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. 2022 Sep 9;17(9):e0273628. doi: 10.1371/journal.pone.0273628

Utilisation and costs of mental health-related service use among adolescents

Carolina Ziebold 1, Wagner Silva-Ribeiro 1,2, Derek King 2, David McDaid 2, Mauricio Scopel Hoffmann 2,3,4,5, Renee Romeo 6, Pedro Mario Pan 1,5, Eurípedes Constantino Miguel 5,7, Rodrigo Affonseca Bressan 1,5, Luis Augusto Rohde 5,8,9, Giovanni Abrahão Salum 5,9, Jair de Jesus Mari 1,5, Sara Evans-Lacko 2,*
Editor: Dylan A Mordaunt10
PMCID: PMC9462733  PMID: 36084089

Abstract

Background

The high level of care needs for adolescents with mental health conditions represents a challenge to the public sector, especially in low and middle-income countries. We estimated the costs to the public purse of health, education, criminal justice and social care service use associated with psychiatric conditions among adolescents in Brazil; and examined whether the trajectory of psychopathology and its impact on daily life, and parental stigma towards mental illness, was associated with service utilisation and costs.

Methods

Data on reported service use among adolescents from a prospective community cohort (n = 1,400) were combined with Brazilian unit costs. Logistic regression and generalised linear models were used to examine factors associated with service use and associated costs, respectively.

Results

Twenty-two percent of those who presented with a psychiatric disorder used some type of service for their mental health in the previous twelve months. Higher odds of service use were associated with having a diagnosed mental disorder (either incident, [OR = 2.49, 95%CI = 1.44–4.30, p = 0.001], remittent [OR = 2.16, 95%CI = 1.27–3.69, p = 0.005] or persistent [OR = 3.01, 95%CI = 1.69–5.36, p<0.001]), higher impact of symptoms on adolescent’s life (OR = 1.32, 95%CI = 1.19–1.47, p<0.001) and lower parental stigma toward mental illness (OR = 1.12, 95%CI = 1.05–1.20, p = 0.001). Average annual cost of service use was 527.14 USD (s.d. = 908.10). Higher cost was associated with higher disorder impact (β = 0.25, 95%CI = 0.12–0.39, p<0.001), lower parental stigma (β = 0.12, 95%CI = 0.02–0.23, p = 0.020) and white ethnicity (β = 0.55, 95%CI = 0.04–1.07, p = 0.036).

Conclusion

The impact of mental health problems on adolescents’ daily lives and parental stigmatising attitudes toward mental illness were the main predictors of both service use and costs.

Introduction

Mental health conditions affect 13.4% of children and adolescents globally, representing the leading cause of disability in this age group [1]. They can have long-term impacts on health and social outcomes into adulthood [27]. The high prevalence and potentially enduring nature of these impacts make addressing youth mental health conditions particularly important, but this is a challenge for public systems with limited resources [8]. Economic costs associated with youth mental health conditions involve a wide range of sectors including health, educational, social care, and criminal justice services [9, 10]. This can represent a substantial cost to the public purse, yet it could also be considered a wise investment given the evidence that effective treatment can mitigate the impact of poor mental health [2]. Estimating the economic cost of mental disorders in young people from the perspective of the public purse and understanding which factors are associated with these costs could support more effective and efficient policy planning and care delivery [8, 11, 12].

Some studies from high–income countries about mental health-related service use among young people suggest that lower socioeconomic status, as well as clinical features (illness severity and impact of disorders) increase the likelihood of service usage in the health, special education, and social care sectors, while male gender and older age are associated with more criminal justice services contacts [11, 13, 14]. These sociodemographic and clinical characteristics are also associated with greater mental health-related treatment costs among young people [11, 14, 15]. Stigma can also influence help-seeking. Research in adult populations show clear links between stigma and reduced help-seeking [16], reduced adherence to treatment and early withdrawal from services [16, 17]. Additionally, stigma is one of the most reported barriers to help-seeking amongst adolescents [18]. Families also play a central role in young people’s contact with services. However, there is limited research about how parental stigma could impact on service use and costs. Stigmatising attitudes toward mental illness amongst parents may influence service contacts due to shame and fear of labelling their youth’s mental health condition [19]. We know of two papers from the UK (using the same data), where lower mental illness-related stigma among caregivers (n = 407), was associated with an increased likelihood of young people’s mental health service use [19], though it was not associated with costs [20]. We know of no studies on the association between mental illness-related stigma among caregivers and young people’s service use and costs outside the UK or in low and middle-income countries (LMICs), where families and young people may face different types of barriers to mental health care. Additionally, most costing studies have focused on a single disorder, commonly autism, attention deficit hyperactivity disorder or conduct disorders [10], and little is known about how, in addition to the type of disorder, whether persistence of psychopathology from childhood to adolescence, disorders’ impact on adolescent’s daily life (i.e. functioning), and key barriers to care such as stigma, could influence costs.

There are a limited number of studies reporting on prevalence of mental health service use in LMICs [2123], however, none use validated service use measures. Moreover, prevalence of any use does not capture the intensity of use (e.g. number or type of visits) needed to understand the economic impact of child mental health problems. From a global mental health perspective, examining this issue in a LMIC context, where resources are scarce, is of major significance. Brazil provides universal access to health services and education for the entire population that is free at the point of use, while private health care and education are used by about 20% of the population [2426]. Estimating the economic cost of mental disorders among young people to the public purse, and understanding which factors are associated with these costs in Brazil is essential for public policy planning, specifically to optimise investment. This approach could also be of value for similar health and welfare systems. Furthermore, examining the variation in costs according to clinical characteristics of adolescents, beyond type of diagnosis, is important as the impact of psychopathology on daily life and the trajectory of psychopathology from childhood to adolescence, may support service planning and resource allocation in relation to clinical characteristics in a preventive and responsive way.

The aim of this study is to estimate the costs associated with health, education, criminal justice and social care services among a cohort of young people in Brazil. We first present the annual aggregate cost to the public purse and then disaggregate this impact to reflect and understand the relative costs to different sectors. Second, we examine how costs vary according to: mental health trajectories, impact of the disorder on everyday life, and parent/guardian stigma towards mental illness. We hypothesise that persistence of psychiatric disorders from childhood to adolescence and associated impact on adolescents’ lives have the greatest influence on costs. However, we also expect that lower levels of parental stigma towards mental illness will predict greater likelihood of service use and hence higher costs.

Methods

Data and participants

This study is nested within the Brazilian High-Risk Cohort (BHRC), which is an ongoing prospective longitudinal study that comprises a community sample and a high‐risk sub‐sample (a sample at increased risk of mental disorders) of young people from Sao Paulo and Porto Alegre, Brazil. A detailed description of the sample and procedures can be found elsewhere [27]. Briefly, during the registry day, 12,500 parents of young people aged 6 to 14 years attending 57 schools (22 in Porto Alegre and 35 in São Paulo) were invited to a screening of mental health disorders using the Family History Screen (FHS) [28]. A total of 8,012 families (9,937 eligible children, 45,394 family members) were interviewed. Based on the percentage of members in the family that screened positively for psychiatric disorders, an index of family load for each potential eligible child was computed. The final cohort comprised 2,511 young people; 957 were randomly selected, and 1,554 were a sub‐sample at increased risk of mental disorders based on the FHS. Cohort participants were interviewed at baseline (aged 6–14 years, calendar year:2010–2011, n = 2,511), and at first follow-up (n = 2010, aged 9–17 years, calendar year 2014, 80% retention rate). Due to an administrative error, the service use questions were not included in the interview schedule for the first 129 participants (6%) of the BHRC first follow-up. Therefore, we were only able to contact a subsample of those who participated at the first follow-up (94%, n = 1,881) to respond to a supplementary interview which included a comprehensive assessment of mental health-related service use (calendar year: 2014–2015, young people participants aged 10–18 years). Among those contacted, 1,400 (74.4%) guardians (in 93.1% of cases the biological mother) completed the interview, 982 (70.1%) by telephone and 418 (29.9%) face-to-face (See flow chart in S1 Fig). There were no significant differences in persistence of psychopathology or impact of psychopathology on adolescents’ lives among respondents versus non-respondents.

This research was carried out in accordance with the latest version of the Declaration of Helsinki. Parental written informed consent was obtained from all the research subjects. Young people provided verbally informed assent (documented as part of the consent form, and witnessed by the interviewer), and those who were able to read and write also provided written consent. All procedures were approved by the Ethics Committee of the Federal University of São Paulo-UNIFESP (N° 2.879.533 and CAAE 06457219.9.0000.5505), Hospital de Clínicas de Porto Alegre (CAAE 06457219.9.3001.5327) and the European Research Commission. Data were provided by the Brazilian High-Risk Cohort study and are available upon request in the Open Science Framework public repository (https://osf.io/ktz5h/).

Measures

Sociodemographic characteristics

Data on the following sociodemographic characteristics were collected: gender, age at follow-up, ethnicity (white and non-white: black, Asian, indigenous or mixed-race), socioeconomic group (SEG), and maternal educational level (no/basic, secondary or university education). SEG was defined according to a Brazilian standardized questionnaire [29]. Based on families’ assets and head of household’s education level, a total score ranging for 0 to 46 is given, where greater scores represent higher socioeconomic status. In this study, SEG was categorised as “low” (0–22) and “high” (23–46).

Psychopathology

Psychiatric diagnosis. Psychiatric diagnoses were assessed at baseline and follow-up using the Brazilian-Portuguese version of the Development and Well-being Assessment (DAWBA) [30, 31], which is a highly structured interview used to generate DSM-IV diagnoses. Trained interviewers gathered information on current problems causing significant distress or social impairment. At baseline, diagnostic assessment and interviews were performed with guardians only. Previous literature has found that self-report on internalising conditions during adolescence is higher compared with parental report. This can be explained because internalising problems, such as anxiety or depression, would be less observable by guardians, being advisable to consider both reports to reach a reliable evaluation of adolescent mental health [32, 33]. For this reason, diagnostic assessment at 3-year follow-up was performed considering guardian reports and additional information from interviews with the young people about internalising conditions. Computerised diagnostic probabilities were then generated based on responses those were carefully evaluated by 9 trained psychiatrists who determined the diagnosis.

Broad psychiatric diagnostic categories. Based on previous literature [34], follow-up DAWBA diagnoses were grouped into three broad categories: distress-related disorders (including depression, generalised anxiety disorder, obsessive–compulsive disorder, tic, eating disorder), fear-related disorders (including panic, agoraphobia, social anxiety, specific phobia and separation anxiety) and externalising disorders (including conduct disorder, oppositional defiant disorder and attention deficit/hyperactivity disorder).

Persistence of diagnosis. Four categories of diagnostic persistence were created based on presence of diagnosis at baseline and/or follow-up: 1) no diagnosis (no diagnosis at both time points), 2) incident (no diagnosis at baseline and presence of diagnosis at follow-up), 3) remittent (presence of diagnosis at baseline and no diagnosis at follow-up), 4) persistent (presence of diagnosis at both time points).

Impact of mental health problems at follow-up. was measured according to the ‘impact supplement’ of the Strength and Difficulties Questionnaire (SDQ) which is part of DAWBA. This supplement assesses the impact of behavioural and emotional difficulties on adolescent’s lives according to guardian reports. A total score (0–10) was generated by summing 5 items: distress, social impairment in family life, friendships, learning, and leisure activities [35]. Higher scores represent greater impact. The impact score has demonstrated internal consistency, cross-informant correlations, and stability measured across time [35].

Parent-reported stigma towards mental health problems

To assess parental stigma, we applied the Brazilian Portuguese version of the Reported and Intended Behaviour Scale (RIBS-BP) [36, 37]. The intended behaviour subscale assesses future intended stigmatising behaviour across four domains: living with, working with, living nearby and continuing a relationship with someone with a mental health problem. Higher scores represent lower stigma. The RIBS-BP has demonstrated good internal consistency, and good to excellent construct validity [37].

Service use

The Service Assessment for Children and Adolescents (SACA) [38] was used to ask guardians about service contacts made in the past 12 months in response to concerns regarding their child’s emotions and behaviour, including alcohol and drugs. The SACA assesses type, nature, frequency and duration of services used, treatments received and settings in which services were delivered. Overall concordance between parent report and records (kappa = 0.76) [38] and test-retest reliability for 12-month (kappa = 0.75–0.86) service use on the parent version of the SACA is strong [39].

We received permission from the SACA developers to translate and adapt the instrument to the Brazilian context in consultation with experts in the Brazilian mental health system to ensure we covered the relevant service types and settings in Brazil. The list of services and professionals was grouped into three sectors: 1) health care: inpatient services (psychiatric hospital, psychiatric unit in a general hospital, alcohol and drug clinic); outpatient services (Centre for psychosocial care [CAPS], which are the community mental health services in Brazil; mental health clinics; specialist mental health professionals (psychiatrists and psychologists in settings other than CAPS and mental health clinics); general health services and professionals (emergency room, paediatrician, general practitioner [GP] or family doctor); 2) education: special school and special education in regular school (special room and special needs class assistant); 3) social care and criminal justice: overnight stay in a shelter or detention centre; probation programme contact; and home visit of the guardianship council (services responsible for child-rights protection).

Estimation of costs

Data collected on use of services from the BHRC were combined with unit costs to derive service use costs in Brazilian Reals for the financial year 2018 and then converted to US dollars (based on December 31 2018 conversion rate 1 Real = 0.2581 dollars, according to the Brazilian Central Bank) [40].

Unit costs. Detailed information on source of information and unit cost values for each service is available in S1 Table. Where available, we applied unit costs previously reported in the Brazilian literature [41, 42]. However, as costs of many services have not previously been reported, we performed a thorough consultation process gathering relevant data from public databases of the Ministries of Education and Health, and the social care departments of the municipalities of Porto Alegre and São Paulo (S1 Table).

Unit costs were attached to data on service use frequencies for each type of service (based on the SACA) based on 2018 prices or the latest available year converted to 2018 prices using the Nationwide Consumer Price Index. The Brazil Central Bank’s calculator was used to apply the index [43]. Once obtained, information on the unit cost of each service was used to calculate the total annual cost by sector (health, education, social care and criminal justice) for each participant by multiplying the frequency of use (e.g. number of visits, nights) by unit cost.

Data analysis

Data were analysed using STATA, version 14. First, we described prevalence of socio-demographic and clinical characteristics overall and by persistence of psychopathology. Between-group differences were compared using chi-squared tests. For interval variables, means and standard deviations were calculated and overall significance was tested using one-way analysis of variance. A significance parameter of p < .05 (two-tailed) was applied for all tests.

Unadjusted odds ratios and coefficients for each predictor and covariate in relation to mental health service use and costs are presented in S2 and S3 Tables. To compare the relative impact between our three main predictors (i.e., psychopathological trajectories, impact of the disorder and parental stigma) of service use and costs we also present logistic regression models for each of these variables adjusting for sociodemographic characteristics (gender, age, mother’s education, ethnicity and SEG) and dummy variables (mode of data collection and city of residence) (S4S6 Tables for service use and S7S9 Tables for costs). We then used multivariable analyses to examine the association between guardian and adolescent characteristics with service use (logistic regression models) and associated costs (generalised linear models–GLM), overall and by sector: 1) health; 2) education; and 3) social care and criminal justice. All multivariable analyses were adjusted by socio-demographic characteristics, mode of data collection and city. For costs GLM, we analysed the subset of participants who used services in the previous 12 months (n = 143). Annual costs for each participant were included in the models as a scalar dependent variable, with a Gamma distribution [44], using the log-link function.

Results

Table 1 describes sociodemographic and clinical characteristics of participants. The sample comprised 1,400 adolescents with a mean age of 14.51 years (s.d = 1.98). The majority were white males from low SEG, and only 10.6% of mothers had university education. 23.3% (n = 326) of adolescents had a psychiatric disorder in the previous 12 months, of which 177 (54.3%) were incident and 149 (45.7%) persistent cases since baseline. 213 (15.2%) participants had remitted from a baseline psychiatric diagnosis. 73 (22.4%) of those who presented with a psychiatric disorder (32 incident and 41 persistent cases) reported using some type of service for their mental health in the previous twelve months. The proportion of service use among those who presented a persistent psychiatric condition was 27.5%. Unadjusted odds ratios of any service use among participants with persistent diagnosis were 7.22 (95%CI = 4.50–11.58, p<0.001) compared with participants with no diagnosis, OR = 1.72 (95%CI = 1.02–2.91, p = 0.043) compared with incident and OR = 2.62 (95%CI = 1.52–4.49, p<0.001) with remittent diagnosis. Table 1 present mean disorder impact by psychiatric trajectories. Unadjusted generalised regression models showed that persistent cases presented greater mean difference in disorder impact (SDQ scores) by 2.34, (95%CI = 2.11–2.58, p<0.001) compared with no diagnosis, 1.14 (95% CI = 0.85–1.42, p<0.001), compared with incident cases, and 1.84 (95% CI = 1.56–2.12, p<0.001), compared with remittent cases. Table 1 also describes the mean costs of mental health-related service use in the past year, by psychiatric trajectory (from no diagnosis to persistent psychiatric diagnosis). Bivariate analyses showed a non-significant association between psychiatric trajectory and mean annual costs.

Table 1. Sociodemographic and clinical characteristics by trajectories of psychopathology (n = 1,400).

No psychiatric diagnosis (n = 861) Incident psychiatric diagnosis (n = 177) Remittent psychiatric diagnosis (n = 213) Persistent psychiatric diagnosis (n = 149) Overall sample (n = 1,400)
N (%) N (%) N (%) N (%) N (%) p
Sociodemographic characteristics
Male gender 503 (58.4) 81 (45.8) 134 (62.9) 83 (55.7) 801 (57.2) 0.005
Female gender 358 (41.6) 96 (54.2) 79 (37.1) 66 (44.3) 599 (42.8)
Age, mean (s.d) 14.50 (2.02) 14.58 (1.90) 14.39 (1.88) 14.67 (1.99) 14.51 (1.98) 0.564
High SEG 359 (41.7) 63 (35.6) 71 (33.3) 61 (40.9) 554 (39.6) 0.095
Low SEG 502 (58.3) 114 (64.4) 142 (66.7) 88 (59.1) 846 (60.4)
White ethnicity 484 (56.2) 106 (60.2) 116 (54.5) 84 (57.1) 790 (56.6) 0.704
Non-White ethnicity 377 (43.8) 70 (39.8) 97 (45.5) 63 (42.9) 607 (43.5)
Guardians characteristics
Maternal no/basic education 387 (45.1) 78 (44.6) 96 (45.3) 59 (39.9) 620 (44.5) 0.953
Maternal secondary education 384 (44.8) 78 (44.6) 93 (43.9) 71 (48.0) 626 (44.9)
Maternal university education 87 (10.14) 19 (10.9) 23 (10.9) 18 (12.2) 147 (10.6)
Clinical characteristics
Any Psychiatric Diagnosis - 177 (54.3) - 149 (45.7) 326 (23.3) <0.001
Fear-related - 92 (52.0) - 72 (48.3) 164 (11.7) <0.001
Distress-related - 70 (40.0) - 60 (40.3) 130 (9.3) <0.001
Externalising - 49 (27.7) - 68 (45.6) 117 (8.4) 0.001
SDQ impact mean score (s.d) 0.28 (0.73) 1.49 (1.91) 0.78 (1.51) 2.62 (2.41) 0.78 (1.52) <0.001
Mental health-related service use
12-months service use 43 (5.0) 32 (18.0) 27 (12.7) 41 (27.5) 143 (10.2) <0.001
Mean service use costs USD$ (s.d) 326.41 (395.53) 581.90 (1360.19) 644.35 (795.50) 628.50 (901.02) 527.14 (908.10) 0.400

Notes: Results in bold are significant. SEG, socioeconomic group; SDQ, Strength and Difficulties Questionnaire. 3 missing data in ethnicity variable, 10 missing data in maternal education variable.

Frequency of mental health-related service use and annual service use costs

Utilisation of mental health services in the previous 12 months and associated cost by type of service are presented in Table 2. Overall, 10.2% of the sample (n = 143) used some sort of health, education, criminal justice or social care service for mental health problems. Disaggregating by sectors, the health sector had highest proportion of service users (9%), while the education and social care and criminal justice sectors were less frequently contacted with a 1.8% and 1.3% of users respectively. Within the heath sector, the outpatient mental health services, most notably psychologists and psychiatrists in settings other than community mental health clinics, were the most frequently used services/professionals. Inpatient services and general health services such as GP/family doctor, paediatrician and emergency department, were less frequently used. In the education sector, school assistant was the most type of service used by young people, while guardianship council was the most frequently social care service contacted. The total cost of 12-month mental health-related service use for the public purse was 70,110.23 USD. The sector that presented higher total annual cost was the health sector, followed by the education and finally the social care and criminal justice sectors. The services that generated the greatest total costs for the heath sector were psychologist (11,339.64 USD) and CAPS (9,628.01 USD). Among those who used services, the average annual cost of service use amounted to 527.14 USD (SD = 908.10 USD, range = 8.77–7,605.58 USD, median = 221.10 USD, interquartile range = 545.28) per user. Individuals using CAPS (specialty mental health) services (1.1% of the sample) had the highest mean number of visits per person during the previous year and the highest associated costs among health services. The second highest mean costs in the health sector were related to hospitalizations in psychiatric hospitals and alcohol and drugs clinics, while the lowest mean costs were attributed to emergency department, paediatrician, outpatient alcohol and drugs and GP/family doctor contacts. Although only 0.1% of individuals used shelters, this type of social service had the highest associated mean cost. Education services were used by 1.8% of individuals and these services had the second highest associated mean costs.

Table 2. 12-month mental health-related service use and costs by type of service (n = 143).

Users Number of visits/ nights Number of nights/visits per usera Total annual cost per service Annual cost per user
Type of service n (%) Total Mean (Range; s.d.) USDa,b Mean (Range; s.d.)
Health Sector
Inpatient mental health services 3
Psychiatric hospital 7 (0.5) 73 10.6 (1–30;11.87) 4,015.72 573.67 (66.42–1,992.72;691.76)
Psychiatric unit in general hospital 1 (0.1) 1 1 (1) 40.49 40.49
AD clinic 3 (0.2) 48 16 (6–27;10.73) 1,767.90 589.30 (191.87–1,096.36;462.10)
Outpatient mental health services
Centre for psychosocial care (CAPS) 15 (1.1) 452 30.15(1–180;49.38) 9,628.01 740.62 (24.56–4,421.03;1212.73)
Mental Health clinic 17 (1.2) 308 18.13(1–70;19.08) 5,644.64 352.79 (19.46–1,362.50;371.28)
Psychiatrist 33 (2.4) 217 6.56 (1–48;8.95) 5,803.90 181.37(27.64–1,326.60;247.29)
Psychologist 71 (5.1) 1,081 15.23 (1–60;14.97) 11,339.64 171.81(11.28–676.99;168.91)
AD clinic 2 (0.1) 2 1 (1) 14.74 14.74
General Health
Emergency department 4 (0.3) 9 2.25 (1–4;1.50) 156.0 39.00 (17.34–69.34;26.00)
Paediatrician 3 (0.2) 10 3.33 (2–4;1.16) 120.54 40.18 (24.11–48.22;13.92)
GP/family doctor 5 (0.4) 23 4.60 (2–9;2.97) 403.25 80.65 (35.07–157.80;52.01)
Overall health service use 126 (9.0) 37,679.94 324.83 (11.28–4575.70;590.55)
Educational sector4
Special School 7(0.5) School Year 8,564.92 1,223.56 (1,155.72–1,250.70; 44.53)
Special Class 5 (0.4) School Year 6,063.55 1,212.71 (1,155.72–1,250.70; 52.02)
School Assistant 12 (0.9) School Year 14,723.52 1,226.96 (1,155.72–1,250.70; 42.95)
Overall education service use 23 (1.8) 29,351.94 1,276.17 (1,155.72–2,501.40; 270.73)
Social care and criminal justice sector
Shelter 2 (0.1) 210 105 (90–120;21.21) 5,599.95 2,799.98 (2,755.34–2,888.48; 63,12)
Guardianship Council home visit 11 (0.8) 31 2.85 (1–5;1.73) 201.84 25.23 (8.77–43.87;15.15)
Probation programme 8 (0.6) Six months 1,875.48 234.44
Overall social care and criminal justice related service use 18 (1.3) 4,687.47 334.82 (8.77–2,888.48;1,155.72)

aTotal cost health sector N = 116, Total cost education sector, N = 23, Total cost social care and criminal justice sector, N = 14. Total cost, N = 133. Cases with missing values in ‘frequency of visits’ were not included in costs estimates: CAPS = 2, mental health clinic = 1, psychiatrist = 1, psychologist = 5, AD clinic = 1, guardianship council = 3.

bCosts are expressed U.S. Dollars, 2018 prices. Brazilian Central Bank conversion rate: Brazilian Real = 0.2581, December 31st 2018[40]

Characteristics associated with mental health-related service use

Having an incident, remittent or persistent psychiatric disorder, as well as the higher impact of behavioural and emotional difficulties on the adolescents’ lives and lower parental stigma, all predicted higher odds of any 12-month service use in unadjusted analyses (S2 Table), in models adjusted by sociodemographic characteristics (S4S6 Tables) and multivariable analyses (Table 3). Service contacts in the health sector were also predicted by the same factors. Service use in the educational sector was predicted by diagnosis trajectory, impact and lower stigma in the unadjusted analyses (S2 Table), and in the models adjusted by sociodemographic characteristics (S4S6 Tables). However, disorder persistence did not remain significant in multivariable analyses, where impact, lower parental stigma and low SEG showed a significant association with education service use (Table 3). Although persistence of the disorder and higher impact of behavioural and emotional difficulties on the adolescents’ lives were associated with social care and criminal justice service use in the unadjusted analyses (S2 Table), and in the models adjusted by sociodemographic characteristics (S4 and S5 Table), there were no factors significantly associated with use of social care and criminal justice services in multivariable analyses (Table 3).

Table 3. Multivariable logistic regression models: Predictors of 12-month mental health service utilisation (n = 1,390a).

Predictors Any service use Health service use Education service use Social care and criminal justice service use
AOR (95%CI) p AOR (95%CI) p AOR (95%CI) p AOR (95%CI) p
Sociodemographic characteristics
Male gender (Ref) - - - -
Female gender 0.87 (0.59–1.28) 0.488 0.98 (0.65–1.46) 0.901 0.67 (0.24-.84) 0.435 1.28 (0.48–3.40) 0.627
Age (in years) 1.02 (0.92–1.12) 0.703 1.00 (0.90–1.11) 0.942 0.98 (0.78–1.25) 0.886 1.25 (0.97–1.60) 0.084
High SEG (Ref)
Low SEG 1.30 (0.86–1.98) 0.211 1.11 (0.72–1.70) 0.646 4.31 (1.29–14.39) 0.018 2.97 (0.75–11.77) 0.122
White ethnicity (Ref) - - - -
Non-White ethnicity 1.17 (0.79–1.72) 0.442 0.97 (0.64–1.46) 0.869 0.53 (0.19–1.48) 0.225 2.59 (0.92–7.28) 0.071
Guardians characteristics
Maternal no/basic education (Ref) - - - -
Maternal secondary education 1.23 (0.82–1.85) 0.315 1.30 (0.84–1.99) 0.238 1.77 (0.66–4.78) 0.257 0.51 (0.17–1.54) 0.233
Maternal university education 1.14 (0.59–2.20) 0.698 1.17 (0.59–2.31) 0.658 1.33 (0.24–7.53) 0.744 1.02 (0.19–5.54) 0.981
Lower parental stigma (RIBS scores) 1.12 (1.05–1.20) 0.001 1.11 (1.03–1.18) 0.003 1.22 (1.01–1.48) 0.042 1.01 (0.94–1.25) 0.251
Clinical characteristics
No psychiatric diagnosis (Ref) - - - -
Incident psychiatric diagnosis 2.49 (1.44–4.30) 0.001 2.57 (1.45–4.58) 0.001 2.29 (0.51–10.97) 0.281 2.54 (0.61–10.52) 0.199
Remittent psychiatric diagnosis 2.16 (1.27–3.69) 0.005 2.22 (1.25–3.93) 0.006 3.24 (0.84–12.50) 0.087 1.98 (0.45–8.75) 0.369
Persistent psychiatric diagnosis 3.01 (1.69–5.36) <0.001 3.33 (1.82–6.08) <0.001 2.82 (0.65–12.37) 0.168 3.65 (0.88–15.09) 0.073
SDQ impact score 1.32 (1.19–1.47) <0.001 1.32 (1.19–1.47) <0.001 1.51 (1.24–1.84) <0.001 1.22 (0.97–1.55) 0.096
Test statistics LR x2(13) = 129.35 p<0.001 LR x2(13) = 122.81, p<0.001 LR x2(13) = 57.46, p<0.001 LR x2(13) = 28.36, p = 0.008
Pseudo-R2 = 0.14 Pseudo-R2 = 0.15 Pseudo-R2 = 0.25 Pseudo-R2 = 0.15

aFrom the total sample, N = 1,400, 10 cases had missing data in mother’s education and 3 in ethnicity variables. AOR = Adjusted odds ratios. Results in bold are statistically significant (p<0.05). Models adjusted by collection instrument and city.

Characteristics associated with greater mental health related service use costs

When all three sectors were combined into a single total cost variable, greater impact and lower parental stigma were associated with higher costs in unadjusted analyses (S3 Table), in the models adjusted by sociodemographic characteristics (S8 and S9 Tables) and multivariable analyses (Table 4). White ethnicity was also associated with higher costs in multivariable analyses (Table 4). Each additional impact score predicted an increase in mean costs of 142.59 USD (p<0.001). For parental stigma, each additional RIBS-BP score (indicating lower stigma) increased mean cost by 69.32 USD (p = 0.020). White ethnicity was associated with having higher mean costs of 295.49 USD (p = 0.036), compared with non-white participants. No association was found between broad diagnosis categories and costs (S10 Table).

Table 4. Generalised linear models: Parental and clinical characteristics associated with cost of mental health service use in the last 12 months, overall and by sector.

Predictors Any service use N = 131 Health service use N = 115 Education service use N = 22 Social care and criminal justice service use N = 14
β (95%CI) p β (95%CI) p β (95%CI) p β (95%CI) p
Sociodemographic characteristics
Male gender (Ref) - - - -
Female gender 0.05 (-0.50–0.59) 0.866 0.06 (-0.61–0.73) 0.854 -0.03 (-0.29–0.24) 0.857 14.41 (-5.34–34.17) 0.153
Age (in years) -0.05 (-0.19–0.10) 0.522 0.06 (-0.14–0.24) 0.572 0.02 (-0.02–0.06) 0.886 -2.11 (-4.87–0.65) 0.133
High SEG (Ref)
Low SEG 0.47 (-0.08–1.03) 0.092 -0.13 (-0.78–0.53) 0.706 -0.03 (-0.36–0.29) 0.839 3.32 (-8.52–15.15) 0.583
White ethnicity (Ref) - - - -
Non-White ethnicity -0.55 (-1.07- -0.04) 0.036 -0.12 (-0.75–0.51) 0.707 0.09 (-0.10–0.27) 0.368 -4.28 (-9.80–1.24) 0.129
Guardians characteristics
Maternal no/basic education (Ref) - - - -
Maternal secondary education 0.27 (-0.29–0.82) 0.341 -0.10 (-0.78–0.58) 0.776 -0.07 (-0.23–0.10) 0.418 4.95 (-2.19–12.08) 0.174
Maternal university education 0.003 (-0.90–0.91) 0.995 -0.34 (-1.38–0.69) 0.515 0.40 (-0.07–0.87) 0.094 - -
Lower parental stigma (RIBS score) 0.12 (0.12–0.39) 0.020 0.04 (-0.07–0.16) 0.465 0.002 (-0.06–0.06) 0.948 0.05 (-0.98–1.08) 0.922
Clinical characteristics
No psychiatric diagnosis (Ref) - - - -
Incident psychiatric diagnosis -0.14 (-0.83–0.55) 0.693 0.15 (-0.71–1.00) 0.735 0.07 (-0.15–0.29) 0.548 -23.61 (-54.48–7.27) 0.134
Remittent psychiatric diagnosis 0.39 (-0.35–1.14) 0.298 0.09 (-0.85–1.04) 0.847 -0.01 (-0.23–0.21) 0.928 -2.17 (-7.42–3.07) 0.417
Persistent psychiatric diagnosis -0.39 (-1.16–0.38) 0.315 -0.42 (-1.40–0.58) 0.412 0.14 (-0.11–0.39) 0.276 -17.23 (-36.84–2.39) 0.085
SDQ impact score 0.25 (0.12–0.39) <0.001 0.20 (1.19–1.47) 0.019 0.01 (-0.02–0.04) 0.458 -0.34 (-1.51–0.83) 0.569
Test statisticsa AIC 16.97193 AIC 16.24671 AIC 20.26365 AIC 15.83608
BIC -353.9633 BIC -308.0671 BIC -24.59063 BIC 1.230216
R2 = 0.22 R2 = 0.15 R2 = 0.79 R2 = 0.90

Notes: Results in bold are significant (p<0.05). Models adjusted by city and method of interview.

aCameron & Windmeijer’s R-squared, measure of goodness of fit for the class of exponential family regression models.

When looking at predictors of costs according to sector (Table 4), disorder impact was associated with greater health sector service use (predicted mean cost by each impact score = 66.26 USD, p = 0.019). We did not find any significant association of psychiatric trajectories, impact of disorder or parental stigma with education sector’s costs (S3, S7S9 Tables, and Table 4). Although persistence of the disorder was associated with social care and criminal justice service’s costs in the unadjusted analyses (S3 Table), and in the models adjusted by sociodemographic characteristics (S7 Table), we did not find any significant factors associated with social care/criminal justice sectors’ costs in multivariable analyses (Table 4).

Discussion

We analysed data on mental health-related service use and associated costs among a prospective community cohort of young people in Brazil. We found that impact of mental health problems on daily life and parental stigma were the most consistent and robust drivers of mental health service use and associated costs.

Drivers of mental-health service use costs

The association between disorder impact and mental health-related service use and costs that we found has been observed in previous research, providing further support that impact and impairment tend to be the strongest and most robust predictors of mental health service use [13, 35] and costs [14]. Contrary to what we expected, we did not find an association between disorder persistence and costs. Our analyses instead found that impact of the disorder on adolescent’s life was the most important clinical predictor and that this was what seemed to drive service costs rather than type or persistence of diagnosis. This finding suggests that although the trajectory of the diagnosis is associated with a greater likelihood of having any contact with mental health-related services, this association did not translate to costs which also reflects the intensity and / or type of care and number of visits per year. Nevertheless, it is important to consider that we have estimated annual costs, and these do not necessarily reflect the cumulative economic costs of persistent cases across childhood and adolescence.

We also found that lower parental stigma was associated with greater service use and higher costs. Our findings suggest that the ways in which parents perceive mental illness in adolescents may significantly influence help-seeking. We are aware of two studies showing that young people’s likelihood of service use across health and education settings was greater among caregivers who reported less intended stigmatising behaviours [19]. Another study indicated that low parental stigmatising attitudes toward mental disorders increased recognition of mental health problems in preadolescents (10–12 years) [45].

We only found one study [20] exploring the impact of parental stigmatising attitudes toward mental illness on child service use costs. The cited study, conducted in the UK, with a smaller sample size, did not find an association between parental stigma and young people’s service use costs. Other research has shown that parental stigma can impede problem recognition and help-seeking [16, 45]. Higher stigma amongst parents and caregivers may discourage or delay service access for their children [19], which may reduce the short-term public sector direct costs of treatment but be detrimental in the long run. Future research needs to further explore the mechanisms through which parental stigma may be related to service/treatment selection and treatment adherence, in order to explain its impact on treatment costs. Moreover, as lower parental stigma may facilitate earlier service contact, it would be interesting to investigate if lower parental stigma may result in lower costs in the longer term.

Among sociodemographic variables, we found that low SEG predicted higher odds of educational service use. This may be related to the fact that young people living in deprived circumstances are more likely to be affected by developmental problems [46], and, therefore, are more likely to use special education services [11]. Although our study did not identify any differences in service use according to ethnicity, we found white ethnicity was associated with higher service use costs. This may reflect disparities in the type of mental health treatment offered or available to non-white children/adolescents. According to previous studies, non-white children/adolescents are less likely to receive adequate mental health treatment [47], including lower likelihood of psychopharmacological prescriptions [48], compared with white children/adolescents.

The economic impact of adolescent mental health care by sectors

We found that the health sector was clearly the main sector accessed by youth with mental disorders. Within the health sector, specialty mental health care was used more frequently and was more costly than primary care. In Brazil, access to CAPS does not require any referral. However, the number of CAPS services are limited, and they are focused on treatment of severe mental disorders [26]. The high costs incurred by the mental health sector for the treatment of psychiatric disorders in CAPS may be because these services provide intensive outpatient treatments (reflected by the highest number of visits we found), which is costly compared with no-specialized services. It is important to highlight that the lack of youth-oriented primary care mental health services in Brazil limits access to treatment. This could explain why we found low frequency of mental health-related contacts with GP/ family doctors. As a result, contact with specialist mental health services only happens when the disorder has significant negative impact on the lives of young people. In this sense, the organisation of a mental health network of care for adolescents, integrating primary care, social care, education, criminal justice and community youth-specialist services, according to the impact of cases, must be considered in Brazil to adequately plan and allocate scarce public budgets [49].

We found that mental-health related educational service use was less prevalent compared with health service use, nevertheless–as previous studies have shown–[11, 14] educational service use was also associated with higher costs. In Brazil, while special education services are provided in regular schools, their use is restricted to students with disabilities and developmental disorders [50], so only adolescents with severe mental disorders are likely to be eligible.

Limitations

Our study has several limitations. First, the psychometric properties of the adapted version of the SACA have not been evaluated yet. Second, as we were not able to access administrative records, service use assessment was limited to guardians’ reports. However, the concordance between parent report and records for service use on the parental version of the SACA is strong [38]. Third, as most of the unit cost were specifically identified for the cities where the BHRC is being conducted, São Paulo and Porto Alegre, they are not necessarily generalisable to the whole country. Fourth, due to the limited number of participants using each type of service, we were unable to compare factors related with use and associated costs of specific types of service. Fifth, due to an administrative error we were unable to contact the first 6% of first follow-up BHRC participants. This could reduce our sample size, but as this was a random error, we do not believe that it affected the results. Furthermore, given our estimates come from observational cohort data, we are not able to establish causality.

Conclusions

Our findings suggest that the main drivers of health-related service use costs among adolescents in Brazil were impact of mental health problems, in addition to lower stigma toward people with mental illness among guardians and White ethnicity. In the present study, only 22.4% of young people with a diagnosed mental disorder received any form of care. In addition to reducing inequality in service use among children, our findings also argue for lowering barriers to care, in particular addressing caregiver stigma. Furthermore, because lower use of services in adolescence may be associated with worse outcomes across the life course [49], further reducing barriers to service utilisation by young people is important, even though this may imply higher short-term costs.

Guardian’s lower stigmatising attitudes towards mental disorders may be crucial to support young people in accessing, engaging and maintaining contact with mental health-related services. Various anti-stigma interventions have demonstrated effectiveness for improving help-seeking [51], but few have been implemented in LMICs. Further studies are needed to design and implement anti-stigma interventions in LMICs. On the other hand, health and education policies need to better support guardians to access appropriate and timely services in their communities, before the symptoms have a significant impact on adolescent functioning. We conclude that the organisation of a culturally sensitive mental health network of care for adolescents, integrating primary care, social care, education, criminal justice services and CAPS, must be considered in Brazil to adequately plan and allocate scarce public budgets.

Supporting information

S1 Fig. Flow chart of Brazilian High-Risk Cohort participants included in the mental-health related service use study.

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S1 Table. Unit costs of health, educational, social care and criminal justice related services.

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S2 Table. Bivariate analysis: Predictors of 12-month mental health service utilisation.

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S3 Table. Bivariate analysis: Predictors of cost of mental health service use in the last 12 months.

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S4 Table. Logistic regression models: 12-month mental health service utilisation predicted by psychiatric diagnosis trajectories.

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S5 Table. Logistic regression models: 12-month mental health service utilisation predicted by impact of behavioural and emotional difficulties on child’ life.

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S6 Table. Logistic regression models: 12-month mental health service utilisation predicted by parental stigma.

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S7 Table. Generalised linear models: Cost of 12-month mental health service utilization predicted by psychiatric diagnosis trajectories.

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S8 Table. Generalised linear models: Cost of 12-month mental health service utilisation predicted by impact of behavioural and emotional difficulties on child’ life.

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S9 Table. Generalised linear models: Cost of 12-month mental health service utilisation predicted by parental stigma.

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S10 Table. Generalised linear models: Cost of 12-month mental health service utilisation predicted by broad diagnosis categories.

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Data Availability

Access to data and Data sharing: CZ have full access to all the data used in the study and takes responsibility for the integrity of the data and the accuracy of the data analysis. Data were provided by the Brazilian High-Risk Cohort study and are available upon request in the Open Science Framework public repository (https://osf.io/ktz5h/).

Funding Statement

The research presented in this article was funded by the European Research Council under the European Union's Seventh Framework Programme (FP7/2007-2013)/ERC grant agreement no 337673, and supported by the UK Medical Research Council (MR/R022763/1), National Institute of Developmental Psychiatry for Children and Adolescents, a science and technology institute funded by Conselho Nacional de Desenvolvimento Científico e Tecnológico (CNPq; National Council for Scientific and Technological Development; grant numbers 573974/2008-0 and 465550/2014-2) and Fundação de Amparo à Pesquisa do Estado de São Paulo (FAPESP grant number 2008/57896-8 and 2014/50917-0). SEL receives support from the UK Medical Research Council in relation to the Mentalkit-Brazil project (MR/R022763/1) and the Economic and Social Research Council. CZ received a doctoral scholarship and research abroad scholarship by the Fundação de Amparo à Pesquisa do Estado de São Paulo (grant number 2018/05586-7 and 2019/08731-0). MSH was supported by the Newton International Fellowship (Ref: NIF\R1\181942), awarded by the Academy of Medical Sciences through the UK Government's Newton Fund Programme. The funding organisations had no role in the study design; collection, analysis or interpretation of data; in the writing of this article; or in the decision to submit the article for publication.

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Decision Letter 0

Therese van Amelsvoort

1 Dec 2021

PONE-D-21-10663Utilisation and costs of mental health-related service use among adolescentsPLOS ONE

Dear Dr. Evans-Lacko,

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Reviewer #1: Yes

Reviewer #2: Yes

**********

2. Has the statistical analysis been performed appropriately and rigorously?

Reviewer #1: Yes

Reviewer #2: Yes

**********

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Reviewer #1: No

Reviewer #2: Yes

**********

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Reviewer #2: Yes

**********

5. Review Comments to the Author

Please use the space provided to explain your answers to the questions above. You may also include additional comments for the author, including concerns about dual publication, research ethics, or publication ethics. (Please upload your review as an attachment if it exceeds 20,000 characters)

Reviewer #1: General

Very relevant and interesting study. Well written paper, I found it pleasant to read. I would recommend some minor adjustments:

Abstract

- When only reading the abstract, the distinction between incident, remittent and persistent disorder in the Results section is a bit confusing. For the abstract, I would recommend rewriting this sentence for example: “Higher odds of service use were associated with having a diagnosed mental disorder (either incident, remittent or persistent), higher impact of symptoms etc.”

Introduction

- This study focuses on the economic cost of mental disorders in young people (line 52). Therefore, it should be better introduced why, in addition to (mental) health services, also education, criminal justice and social care services were investigated.

- Line 55: male gender is mostly not associated with higher use of mental health services. Please specify the association between these factors and specific services.

Methods: Data and participants

- I understand that not all information about the Brazilian High-Risk Cohort was included in this paper. I would want to know, however, based on what information the children became part of this high risk cohort. Are they COPMI?

Methods: Measures

- Why only maternal educational level?

- Furthermore, this paragraph forms a clear description of appropriate measures.

Results

- Very clear description and informative tables.

Discussion

- Line 325: “We found that the health sector was clearly the main sector providing mental health care for youth.” That’s quite obvious. I would recommend rewriting this, for example: “We found that the health sector was clearly the main sector accessed by youth with mental disorders.”

- In the present study, only 20% of young people with a diagnosed mental disorder received any form of care. In addition to reducing inequality in service use among children, these data also argue for lowering barriers to care for young people in general. I would recommend stating this in the conclusion as well.

- Line 329: “The lack of youth-oriented primary care mental health programmes”. Is this also the reason why GP’s/family doctors were less frequently visited?

- Line 359-361: this reads like the impact of mental health problems on children’s lives should be increased because it would support help-seeking. Please, rewrite.

- Line 363: effectives should be effective

- In future research, it would be interesting to not only assess parental stigma but also stigma among the adolescents themselves.

Reviewer #2: It's good to see more representative research from LMICs, trying to bridge the existing knowledge gap. This study's most significant plus point is that it looks at service use and service cost from multiple angles, shedding light on demographic, clinical and systemic factors that contribute to service use cost. However, this manuscript does require significant improvement in language and content. Here are my main suggestions:

1) The language of the manuscript can be crisper. Multiple places sentences look disjointed or elongated. The paragraphs are changed too frequently in some places, with each of these paragraphs containing only one or two sentences.

2) Introduction: In line 57, please clarify whether by 'education services' authors mean remedial education services or some other kind of services?

3) Introduction: The lines 55-58 are difficult to follow: authors claim that certain demographic and clinical characteristics are associated with a greater likelihood of using certain services as per existing research. However, it's not clear how this connects with the assertion about young people in the same sentence.

4) Introduction: The importance of studying parental stigma needs to be built better.

5) Introduction: I'm not sure what is meant by 'beyond diagnosis', are authors implying the existing studies cover the cost of diagnosis only or for limited kinds of disorders. Some clarification here would be helpful.

6) Introduction: The way lines 72-73 are written makes it sound like Brazil is a high-income country

7) Introduction: In line 88, it's unclear what characteristics the authors are referring to and whether the following hypothesis is related to a subset of these characteristics?

8) Methods: In line 96, some information on how these children were classified as high risk will be helpful. The authors have said the details are somewhere else, but a brief description here will make it easier for the reader to understand the sample.

9) Methods: In line 99, it was slightly hard to follow study timelines. Was this study carried out after the first follow-up in 2014-2015 or as part of the follow-up?

10) Methods: The authors can use consistent terminology: children or young people. As of now, this has varied from one sentence to another.

11) the '-' in line 102 seems typo.

12) Methods: In lines 127-130, it's unclear why young people were not interviewed at baseline but were included during the 3-year follow-up?

13) Methods: Do authors have any psychometric properties of the adapted version of Service Assessment for Children and Adolescents that can be reported in this publication?

14) Results: In line 224, the authors refer to Table 1. However, without any commentary on the significance of data in this table, the authors jump to a new set of findings. All this makes it slightly hard to follow what is being presented.

15) The 12-month service use and service use cost means are presented in Tables 1 and 2. Repeating the same findings across two tables should be avoided

16) The paragraph on page 12 lacks a description of the cost associated with each service? For e.g., although CAPS is not a highly prevalent service, the associated cost makes for a lion contribution to the public purse. This needs to be presented and discussed.

17) Discussion: In line 288, the use of the terms 'above and beyond' doesn't convey much. To the best of my knowledge, the current analysis nowhere helps to reach this conclusion of above and beyond. I am requesting authors to look at terminology closely.

18) Discussion: Lines 301-312 can be streamlined and better organised.

19) Discussion: Line 327: The number of CAPS users was less, but the number of visits and costs for those who used it were very high. These were not reflected in the discussion, nor were its implication for the restructuring health system.

20) Discussion: The hypothesis stated that researchers were interested in examining the impact of persistence of psychiatric disorders from childhood to adolescence on service costs; however, the discussion did not give much attention to this part.

21) Conclusion: Some of the text in the last paragraph of the conclusion, i.e. those referring to implications, can be moved to discussion and expanded further.

I'm not able to comment on cost analysis as this is not my area of expertise.

**********

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Reviewer #1: No

Reviewer #2: Yes: KANIKA MALIK

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PLoS One. 2022 Sep 9;17(9):e0273628. doi: 10.1371/journal.pone.0273628.r002

Author response to Decision Letter 0


15 Feb 2022

We appreciate the careful revision of our manuscript and the comments of the reviewers. We are pleased to be invited to submit the revised version of our paper to PLOS ONE.

Please find attached both an unmarked version of the revised manuscript and one version with changes marked in red. Our point-by-point responses to the reviewers’ comments (unquoted italics) and details of the changes we have performed to our revised manuscript are given below.

Reviewer #1:

General Comment: Very relevant and interesting study. Well written paper, I found it pleasant to read. I would recommend some minor adjustments

Response: We appreciate your positive feedback, the careful revision of our manuscript and your comments.

Comment 1: Abstract- When only reading the abstract, the distinction between incident, remittent and persistent disorder in the Results section is a bit confusing. For the abstract, I would recommend rewriting this sentence for example: “Higher odds of service use were associated with having a diagnosed mental disorder (either incident, remittent or persistent), higher impact of symptoms etc.”

Response: Thank you for your comment. We have rewritten this sentence as follows:

Higher odds of service use were associated with having a diagnosed mental disorder (either incident [OR=2.49, 95%CI=1.44-4.30, p=0.001], remittent [OR=2.16, 95%CI=1.27-3.69, p=0.005] or persistent [OR=3.01, 95%CI=1.69-5.36, p<0.001]), higher impact of symptoms..

Comment 2: Introduction- This study focuses on the economic cost of mental disorders in young people (line 52). Therefore, it should be better introduced why, in addition to (mental) health services, also education, criminal justice and social care services were investigated.

Response: We have edited the introduction as follows:

The high prevalence and potentially enduring nature of these impacts make addressing youth mental health conditions particularly important, but this is a challenge for public systems with limited resources (Knapp M; Evans-Lacko S, 2015). Economic costs associated with youth mental health conditions involve a wide range of sectors including health, educational, social care, and criminal justice services [9,10]. This can represent a substantial cost to the public purse, yet it could also be considered a wise investment given the evidence that effective treatment can mitigate the impact of poor mental health (Knapp et al., 2011).

Comment 3: Line 55: male gender is mostly not associated with higher use of mental health services. Please specify the association between these factors and specific services.

Response: We appreciate your suggestion. We have edited this paragraph in the revised version of the manuscript:

Some studies from high–income countries suggest that lower socioeconomic status, as well as clinical features (illness severity and impact of disorders) are associated with use of health, special education, and social care services, while male gender and older age are associated with more criminal justice services contacts [11,13,14]. These sociodemographic and clinical characteristics are also associated with greater mental health-related treatment costs among young people [11,14,15]

Comment 4: Methods: Data and participants. I understand that not all information about the Brazilian High-Risk Cohort was included in this paper. I would want to know, however, based on what information the children became part of this high risk cohort. Are they COPMI?

Response: Thanks for the important point you raised. We have added information in the methods on the Brazilian High-Risk cohort sampling procedures as follows:

This study is nested within the Brazilian High-Risk Cohort (BHRC), which is an ongoing prospective longitudinal study that comprises a community sample and a high‐risk sub‐sample (a sample at increased risk of mental disorders) of young people from Sao Paulo and Porto Alegre, Brazil. A detailed description of the sample and procedures can be found elsewhere [25]. Briefly, during the registry day, 12,500 parents of young people aged 6 to 14 years attending 57 schools (22 in Porto Alegre and 35 in São Paulo) were invited to a screening of mental health disorders using the Family History Screen (FHS) [26]. A total of 8,012 families (9,937 eligible children, 45,394 family members) were interviewed. Based on the percentage of members in the family that screened positively for psychiatric disorders, an index of family load for each potential eligible child was computed. The final cohort comprised 2,511 young people; 957 were randomly selected, and 1,554 were a sub‐sample at increased risk of mental disorders based on the FHS.

Comment 5: Methods: Measures. Why only maternal educational level?

- Furthermore, this paragraph forms a clear description of appropriate measures.

Response: As stated in the methods section, the socioeconomic group variable comprised head of household educational level in addition to other household socioeconomic indicators. As some research suggests that mothers educational level is particularly important for recognition and help-seeking, we also included this variable as a separate indicator. As the vast majority of caregiver respondents were mothers (in 93% of cases the biological mother [information included in the revised manuscript]) we focused on maternal education rather than estimating the educational level of other caregivers.

Comment 6: Results. Very clear description and informative tables.

Response: Thank you very much for your positive feedback.

Comment 7: Discussion- Line 325: “We found that the health sector was clearly the main sector providing mental health care for youth.” That’s quite obvious. I would recommend rewriting this, for example: “We found that the health sector was clearly the main sector accessed by youth with mental disorders.”

Response: Thank you very much for your suggestion. We rewrote this sentence as follows:

We found that the health sector was clearly the main sector accessed by youth with mental disorders.

Comment 8: In the present study, only 20% of young people with a diagnosed mental disorder received any form of care. In addition to reducing inequality in service use among children, these data also argue for lowering barriers to care for young people in general. I would recommend stating this in the conclusion as well.

Response: Thanks for your suggestion. We have edited the first paragraph of the conclusions as follows:

Our findings suggest that the main drivers of health-related service use costs among adolescents in Brazil were impact of mental health problems, in addition to lower stigma toward people with mental illness among guardians and White ethnicity. In the present study, only 22.4% of young people with a diagnosed mental disorder received any form of care. In addition to reducing inequality in service use among children, our findings also argue for lowering barriers to care, in particular addressing caregiver stigma. Furthermore, because lower use of services in adolescence may be associated with worse outcomes across the life course [47], it is needed to further explore measures to reduce inequalities in service utilisation by young people, even though this implies higher short-term costs.

Comment 9: Line 329: “The lack of youth-oriented primary care mental health programmes”. Is this also the reason why GP’s/family doctors were less frequently visited?

Response: We appreciate your comment, and we agree with your interpretation of this result. We have edited the referred sentence:

The lack of youth-oriented primary care mental health programmes limits access to treatment when symptoms start to have an impact on adolescent functioning. This can explain why we found a low rate of mental health-related contacts with GP/ family doctors. As a result, contact with specialist mental health services only happens when the disorder has significant negative impact on the lives of young people.

Comment 10: Line 359-361: this reads like the impact of mental health problems on children’s lives should be increased because it would support help-seeking. Please, rewrite.

Response: We have rewritten this paragraph:

Guardian’s lower stigmatising attitudes towards mental disorders may be crucial to support young people in accessing, engaging and maintaining contact with mental health-related services. Various anti-stigma interventions have demonstrated effectiveness for improving help-seeking [49], but few have been implemented in LMICs. Further studies are needed to design and implement anti-stigma interventions in LMICs. On the other hand, health and education policies need to better support guardians to access appropriate and timely services in their communities, before the symptoms have a significant impact on adolescent functioning.

Comment 11: Line 363: effectives should be effective

Response: Thank you very much, we have corrected this error.

Comment 12: In future research, it would be interesting to not only assess parental stigma but also stigma among the adolescents themselves.

Response: We agree with you, and we are planning to evaluate the association between mental health-related service use and youth stigma towards mental illness in future cohort’s assessments.

Reviewer #2:

General comment: It's good to see more representative research from LMICs, trying to bridge the existing knowledge gap. This study's most significant plus point is that it looks at service use and service cost from multiple angles, shedding light on demographic, clinical and systemic factors that contribute to service use cost. However, this manuscript does require significant improvement in language and content. Here are my main suggestions:

Response: We appreciate your positive opinion of our work, the careful revision of our manuscript and your valuable comments.

Comment 1: The language of the manuscript can be crisper. Multiple places sentences look disjointed or elongated. The paragraphs are changed too frequently in some places, with each of these paragraphs containing only one or two sentences.

Response: Thanks for your comment. We have revised and edited the language through the manuscript.

Comment 2: Introduction: In line 57, please clarify whether by 'education services' authors mean remedial education services or some other kind of services?

Response: Thanks for your comment. We have indicated ‘special education’ in the revised version of the manuscript.

Comment 3: Introduction: The lines 55-58 are difficult to follow: authors claim that certain demographic and clinical characteristics are associated with a greater likelihood of using certain services as per existing research. However, it's not clear how this connects with the assertion about young people in the same sentence.

Response: Thanks for your comment. We have edited and separated these sentences:

Some studies from high–income countries suggest that lower socioeconomic status, as well as clinical features (illness severity and impact of disorders) are associated with use of health, special education, and social care services, while male gender and older age are associated with more criminal justice service contacts [11,13,14]. These sociodemographic and clinical characteristics are also associated with greater mental health-related treatment costs among young people [11,14,15].

Comment 4: Introduction: The importance of studying parental stigma needs to be built better.

Response: We appreciate your suggestion. We have included the following changes:

Families also play a central role in young people’s contact with services. One study from the UK found that lower mental illness-related stigma among caregivers was associated with an increased likelihood of young people’s mental health service use [16]. Stigmatising attitudes toward mental illness amongst parents may influence service contacts due to shame and fears of labelling their child’s mental health condition [16]. There are clear links between stigma and reduced help-seeking [17], reduced adherence to treatment and early withdrawal from services [17,18]. However, little is known about/ how parental stigma could impact on young people service use and costs.

Comment 5: Introduction: I'm not sure what is meant by 'beyond diagnosis', are authors implying the existing studies cover the cost of diagnosis only or for limited kinds of disorders. Some clarification here would be helpful.

Response: Thanks for your suggestion. We have edited this sentence as follows:

Additionally, little is known about how, in addition to the type of disorder, whether persistence of psychopathology from childhood to adolescence, disorders’ impact on adolescent’s daily life (i.e., functioning), and key barriers to care such as stigma, could influence costs.

Comment 6: Introduction: The way lines 72-73 are written makes it sound like Brazil is a high-income country

Response: We appreciate your comment. We have deleted ‘Similar to most high income countries’ in the revised version of the manuscript.

Comment 7: Introduction: In line 88, it's unclear what characteristics the authors are referring to and whether the following hypothesis is related to a subset of these characteristics?

Response: We have rewritten this sentence to clarify the characteristics under study:

Second, we examine how costs vary according to: mental health trajectories, impact of the disorder on everyday life, and parent/guardian stigma towards mental illness.

Comment 8: Methods: In line 96, some information on how these children were classified as high risk will be helpful. The authors have said the details are somewhere else, but a brief description here will make it easier for the reader to understand the sample.

Response: Thanks for your suggestion. As explained in response to Reviewer 1’s comment 4, we have included a brief description of the Brazilian High-Risk Cohort sampling procedures.

Comment 9: Methods: In line 99, it was slightly hard to follow study timelines. Was this study carried out after the first follow-up in 2014-2015 or as part of the follow-up?

Response: We have tried to clarify this including the following information:

Cohort participants were interviewed at baseline (aged 6-14 years, calendar year:2010-2011, n=2,511), and at first follow-up (N=2010, aged 9-17 years, calendar year 2014). After completing the BHRC first follow-up interview, 1,881 parents/guardians were invited to respond to a supplementary interview which included a comprehensive assessment of mental health related service use (calendar year: 2014-2015, young people participants aged 10-18 years).

Comment 10: Methods: The authors can use consistent terminology: children or young people. As of now, this has varied from one sentence to another.

Response: Thanks for your comment. We have revised and edited the methods section in order to use consistently the term young people.

Comment 11: The '-' in line 102 seems typo.

Response: We appreciate your comment. We have deleted this typo.

Comment 12: Methods: In lines 127-130, it's unclear why young people were not interviewed at baseline but were included during the 3-year follow-up?

Response: This was because participants were younger at baseline and so we relied on parent’s report, given limitations in funding and resources. Given that older adolescents are better at reporting internalising symptoms, both guardian and youth interviews were performed at 3-year follow-up. We included this explanation in the revised version of the manuscript:

At baseline, diagnostic assessment and interviews were performed with guardians only. Previous literature has found that self-reports on internalising conditions during adolescence is higher compared with parental report. This can be explained because internalising problems, such as anxiety or depression, would be less observable by guardians, being advisable to consider both reports to reach a reliable evaluation of adolescent mental health [30,31]. For this reason, diagnostic assessment at 3-year follow-up was performed considering guardian reports and additional information from interviews with the young people about internalising conditions.

Comment 13: Methods: Do authors have any psychometric properties of the adapted version of Service Assessment for Children and Adolescents that can be reported in this publication?

Response:

The parent-report SACA has been shown to be a valid measure of young people’s service use (kappa = 0.76; [Hoagwood et al., 2000]) with test-retest reliability for past-year reports (ranging from 0.75 to 0.86; [Horwitz et al., 2001]). We have not assessed the psychometric properties of the adapted version of the Service Assessment for Children and Adolescents for Brazilian participants yet. We have included this limitation in the revised version of the manuscript.

Comment 14: Results: In line 224, the authors refer to Table 1. However, without any commentary on the significance of data in this table, the authors jump to a new set of findings. All this makes it slightly hard to follow what is being presented.

Response: We appreciated your comment. We have edited this paragraph:

Table 1 describes sociodemographic and clinical characteristics of participants. The sample comprised 1,400 adolescents with a mean age of 14 years (s.d=1.98). The majority were white males from low SEG, and only 10% of mothers had university education. 23.3% (n= 326) of adolescents had a psychiatric disorder in the previous 12 months, of which 177 (54.3%) were incident and 149 (45.7%) persistent cases since baseline. 213 (15.2%) participants had remitted from a baseline psychiatric diagnosis. Participants with externalising disorders were more likely to have persistent trajectories (RR=2.19, 95%CI=1.38-3.48, p<0.001). Participants categorised as persistent also reported greater disorder impact (�=2.34, 95%CI=2.11-2.58, p<0.001). 22.4% of those who presented with a psychiatric disorder reported using some type of service for their mental health in the previous twelve months. The proportion of service use among those who presented a persistent psychiatric condition was 27%. Table 1 also describes the mean costs of mental health-related service use in the past year, by psychiatric trajectory (from no diagnosis to persistent psychiatric diagnosis). Bivariate analyses showed a non-significant association between psychiatric trajectory and mean annual costs.

Comment 15: The 12-month service use and service use cost means are presented in Tables 1 and 2. Repeating the same findings across two tables should be avoided

Response: We have deleted the last line of Table 2 (overall services cost).

Comment 16: The paragraph on page 12 lacks a description of the cost associated with each service? For e.g., although CAPS is not a highly prevalent service, the associated cost makes for a lion contribution to the public purse. This needs to be presented and discussed.

Response: Thank you very much for your suggestion. We edited this paragraph as follows:

Utilisation of mental health services in the previous 12 months and associated cost by type of service are presented in Table 2. Overall, 10.0% of the sample (n=143) used some sort of health, education, criminal justice or social care service for mental health problems. Disaggregating by sectors, the health sector had highest proportion of service users (9%), while the education and social care and criminal justice sectors were less frequently contacted with a 1.8% and 1.3% of users, respectively. Outpatient mental health services, most notably psychologists and psychiatrists in settings other than community mental health clinics, were the most frequently used services/professionals. Inpatient services and general health services such as GP/family doctor, paediatrician and emergency department, were less frequently used.

In the education sector, school assistant was the most type of service used by young people, while guardianship council was the most frequently social care service contacted. The total cost of 12-month mental health-related service use for the public purse was 70,110.23 USD. The sector that presented higher total annual cost was the health sector, followed by the education and finally the social care and criminal justice sectors. The services that generated the greatest total costs for the heath sector were psychologist (11,339.64 USD) and CAPS (9,628.01 USD). Among those who used services, the average annual cost of service use amounted to 527.14 USD (SD= 908.10 USD, range=8.77- 7,605.58 USD, median=221.10 USD, interquartile range=545.28) per user. Individuals using CAPS (specialty mental health) services (1.1% of the sample) had the highest mean number of visits during the previous year and the highest associated costs among health services. The second highest mean costs in the health sector were related to hospitalizations in psychiatric hospitals and alcohol and drugs clinics, while the lowest mean costs were attributed to emergency department, paediatrician, outpatient alcohol and drugs and GP/family doctor contacts. Although only 0.1% of individuals used shelters, this type of social service had the highest associated mean cost. Education services were used by 1.8% of individuals and these services had the second highest associated mean costs.

Comment 17: Discussion: In line 288, the use of the terms 'above and beyond' doesn't convey much. To the best of my knowledge, the current analysis nowhere helps to reach this conclusion of above and beyond. I am requesting authors to look at terminology closely.

Response: Thanks for your suggestion. We have removed this language.

We found that impact of mental health problems on daily life and parental stigma were the most consistent and robust drivers of mental health service use and associated costs..

Comment 18: Discussion: Lines 301-312 can be streamlined and better organised.

Response: We have edited the cited lines as follows:

We did not find any study exploring the impact of parental stigmatising attitudes toward mental illness on child treatment costs. Other research has shown that parental stigma can impede problem recognition and help-seeking [17,43]. Higher stigma amongst parents and caregivers may discourage or delay service access for their children [16], which may reduce the short-term public sector direct costs of treatment but be detrimental in the long run. Future research needs to further explore the mechanisms through which parental stigma may be related to service/treatment selection and treatment adherence, in order to explain its impact on treatment costs. Moreover, as lower parental stigma may facilitate earlier service contact, it would be interesting to investigate if lower parental stigma may result in lower costs in the longer term.

Comment 19: Discussion: Line 327: The number of CAPS users was less, but the number of visits and costs for those who used it were very high. These were not reflected in the discussion, nor were its implication for the restructuring health system.

Response: We appreciate your comment. We have edited the discussion as suggested:

In Brazil, access to CAPS does not require any referral. However, the number of CAPS services are limited, and they are focused on treatment of severe mental disorders [24]. The high costs incurred by the mental health sector for the treatment of psychiatric disorders in CAPS may be a result of both, the severity of patients consulting these services and because these services provide intensive outpatient treatments (reflected by the highest number of visits we found), which is costly compared with no-specialized services. It is important to highlight that the lack of youth-oriented primary care mental health services in Brazil which limits access to treatment. This could explain why we found low frequency of mental health-related contacts with GP/ family doctors. As a result, contact with specialist mental health services only happens when the disorder has significant negative impact on the lives of young people. (Moved from the conclusion as suggested in your last comment). In this sense, the organisation of a mental health network of care for adolescents, integrating primary care, social care, education, criminal justice and community youth-specialist services, according to the impact of cases, must be considered in Brazil to adequately plan and allocate scarce public budgets [47].

Comment 20: Discussion: The hypothesis stated that researchers were interested in examining the impact of persistence of psychiatric disorders from childhood to adolescence on service costs; however, the discussion did not give much attention to this part.

Response: Thanks for rising this important comment. We have included the following paragraph:

Contrary to what we expected, we did not find an association between disorder persistence and costs. Our analyses instead found that impact of the disorder on adolescent’s life was the most important clinical predictor and that this was what seemed to drive service use rather than type or persistence of diagnosis. Nevertheless, it is important to consider that we have estimated annual costs, and these do not necessarily reflect the cumulative economic costs of persistent cases across childhood and adolescence.

Comment 21: Conclusion: Some of the text in the last paragraph of the conclusion, i.e. those referring to implications, can be moved to discussion and expanded further.

I'm not able to comment on cost analysis as this is not my area of expertise.

Response: Thanks for your suggestions, we have moved some conclusions to the discussion as explained in response to your Comment #20.

Attachment

Submitted filename: Response to Reviewers.docx

Decision Letter 1

Therese van Amelsvoort

25 Feb 2022

PONE-D-21-10663R1Utilisation and costs of mental health-related service use among adolescentsPLOS ONE

Dear Dr. Evans-Lacko,

Thank you for submitting your manuscript to PLOS ONE. There are still a few minor points that need addressing. Therefore, we invite you to submit a revised version of the manuscript that addresses the points raised during the review process. Please submit your revised manuscript by Apr 11 2022 11:59PM. If you will need more time than this to complete your revisions, please reply to this message or contact the journal office at plosone@plos.org. When you're ready to submit your revision, log on to https://www.editorialmanager.com/pone/ and select the 'Submissions Needing Revision' folder to locate your manuscript file.

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Reviewer #2: It's good to see the quality of the manuscript and the text organisation has been improved. I'm sharing a few follow-up queries. A sincere request to authors to include line number where revisions are reflected on the non-tracked version. Without line numbers, its difficult to locate revisions.

Introduction:

Follow-up query to comment 3: In lines 57-59, the sentence's meaning is still not very clear. Are the mentioned demographic and clinical features associated with the frequent use of these services or the number of services used? Also, are these findings coming from adult literature, youth literature or across the age span?

Follow-up query to comment 4: In lines 65-69, the revised reasoning looks circular. The authors say that stigmatizing attitude among parents was associated with poor help-seeking. But in the following line states that little is known about the impact of parental stigma on help-seeking. Are authors implying that what is known about children is not applicable to adolescents? It's unclear how the existing findings relate to the gap that the author asserts in this para.

The organization of the text in the introduction section can be improved as a couple of paragraphs have just one or two lines.

Methods

In line 121- is this '-' a typo?

In Line 116-121, it is not clear to the reader how the study sample was reduced to 1881 from the original 2251. I'm guessing the authors included a sub-group of individuals meeting the age criteria. This needs to be clearly defined in the manuscript.

Results

Follow-up query to comment 14: It was helpful to see the description of the results in text. A few comments on the revised text: In line 253, the value in the text doesn't match with the value given in the table. If the rounding up was done, the number of decimal places should be consistent across the text. In line 262, there is no description of whether this service use is significantly different from other trajectories?

Follow-up query to comment 16: Again, it was helpful to see the description of the results in text. A few comments on the revised text: In line 276, there should be a full stop before starting the following sentence. Also, it may be good to start as "within the health sector, the outpatient…", so the reader is clear that now you are looking at the frequency of usage within each sector.

In line 289, it might be good to specify the mean visit per person, so it's clear to the reader that the mean refers to the mean visit per person.

The source of data in lines 334-337 is not provided.

There are a lot of supplementary data files, but except for two supplementary files, others were not referenced or explained in the results section. It would be helpful to understand the reason for including all this analysis but not using them in results.

Discussion

The findings discussed in lines 342-346 don't match the result. The service use was different among those with different trajectories. However, the costs were not different among these groups. Does any of the existing analysis carried out by the author can explain why the difference in frequency of use didn't result in differences in cost between these groups.

Follow-up query to comment 18: The revision looks good. However, in line 347, the authors' assertion that these findings are novel seems misleading. As in quick succession, they have indicated that other studies have also found similar findings.

Follow-up query to comment 19: The discussion points on CAPS results in lines 380-385 are difficult to follow. There is no mention in results about the severity of disorders and types of service accessed. So understanding this link in discussion is difficult.

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Attachment

Submitted filename: PONE-D-21-10663_R1_WC.pdf

PLoS One. 2022 Sep 9;17(9):e0273628. doi: 10.1371/journal.pone.0273628.r004

Author response to Decision Letter 1


22 Apr 2022

We appreciate the careful revision of our manuscript and the comments of the reviewers. We are pleased to be invited to submit the revised version of our paper to PLOS ONE.

Please find attached both an unmarked version of the revised manuscript and one version with changes marked in red. Our point-by-point responses to the reviewers’ comments (unquoted italics) and details of the changes we have performed to our revised manuscript are given below.

Reviewer’s general comment: It's good to see the quality of the manuscript and the text organisation has been improved. I'm sharing a few follow-up queries. A sincere request to authors to include line number where revisions are reflected on the non-tracked version. Without line numbers, it’s difficult to locate revisions.

We appreciate your careful revision of our manuscript and all your comments and suggestions. We have included the line number to locate the revisions of our manuscript.

Follow-up query to comment 3: In lines 57-59, the sentence's meaning is still not very clear. Are the mentioned demographic and clinical features associated with the frequent use of these services or the number of services used? Also, are these findings coming from adult literature, youth literature or across the age span?

Thanks for your comment. We have edited this paragraph as follows:

Some studies from high–income countries about mental health-related service use among young people suggest that lower socioeconomic status, as well as clinical features (illness severity and impact of disorders) increase the likelihood of health, special education, and social care services, while male gender and older age are associated with more criminal justice services contacts [11,13,14]. (Introduction, lines 58-60)

Follow-up query to comment 4: In lines 65-69, the revised reasoning looks circular. The authors say that stigmatizing attitude among parents was associated with poor help-seeking. But in the following line states that little is known about the impact of parental stigma on help-seeking. Are authors implying that what is known about children is not applicable to adolescents? It's unclear how the existing findings relate to the gap that the author asserts in this para.

Thanks for your comment. We have asserted this paragraph in order to explain that there is a clear link established between stigma influencing help-seeking among adults and adolescents (we have added one reference on the later topic), however, much less data on parental stigma in relation to their child’s service use has been published. We have added the only study that investigated the association between parental stigma and young people’s service use in the UK, published on January 27th, 2022. This paragraph was edited as follows:

Stigma can also influence help-seeking. Research in adult populations show clear links between stigma and reduced help-seeking [16], reduced adherence to treatment and early withdrawal from services [16,17]. Additionally, stigma is one of the most reported barriers to help-seeking amongst adolescents [18]. Families also play a central role in young people’s contact with services. However, there is limited research about how parental stigma could impact on service use and costs. Stigmatising attitudes toward mental illness amongst parents may influence service contacts due to shame and fear of labelling their youth’s mental health condition [19]. We know of two papers from the UK (using the same data), where lower mental illness-related stigma among caregivers (n=407), was associated with an increased likelihood of young people’s mental health service use [19], though it was not associated with costs [20]. We know of no studies on the association between mental illness-related stigma among caregivers and young people’s service use and costs outside the UK or in low and middle-income countries (LMICs), where families and young people may face different types of barriers to mental health care. (Introduction, lines 64-77)

New References were updated:

18. Aguirre Velasco A, Cruz ISS, Billings J, Jimenez M, Rowe S. What are the barriers, facilitators and interventions targeting help-seeking behaviours for common mental health problems in adolescents? A systematic review. BMC Psychiatry. 2020;20: 293. doi:10.1186/s12888-020-02659-0

20. Ribeiro WS, Romeo R, King D, Owens S, Gronholm PC, Fisher HL, et al. Influence of stigma, sociodemographic and clinical characteristics on mental health-related service use and associated costs among young people in the United Kingdom. Eur Child Adolesc Psychiatry. 2022. doi:10.1007/s00787-022-01947-2

The organization of the text in the introduction section can be improved as a couple of paragraphs have just one or two lines.

Thanks for your suggestion. We have merged two paragraphs: lines 77 and 92.

Methods

In line 121- is this '-' a typo?

We have edited this sentence as follows:

Among those contacted, 1,400 (74.4%) guardians (in 93.1% of cases the biological mother) completed the interview, 982 (70.1%) by telephone and 418 (29.9%) face-to-face (See flow chart in S1 Fig.). (Line 126)

In Line 116-121, it is not clear to the reader how the study sample was reduced to 1881 from the original 2251. I'm guessing the authors included a sub-group of individuals meeting the age criteria. This needs to be clearly defined in the manuscript.

We appreciate your comment. The Brazilian High-Risk Cohort (BHRC) comprises 2511 children, 2010 of them (80%) participated at the first follow-up. Due to an administrative error, we were only able to contact a subsample of those who participated at first follow-up (94%, n=1881) to participate in our additional research and thus we invited 1881 guardians to complete further questions on mental health-related service use and associated barriers to care. Of those participants who we contacted, 1,400 (74%) agreed to participate in our study.

We have included the following changes (in red) in lines 119-128:

Cohort participants were interviewed at baseline (aged 6-14 years, calendar year: 2010-2011, n=2,511), and at first follow-up (n=2010, aged 9-17 years, calendar year 2014, 80% retention rate). Due to an administrative error, we were only able to contact a subsample of those who participated at the first follow-up (94%, n=1,881) to respond to a supplementary interview which included a comprehensive assessment of mental health-related service use (calendar year: 2014-2015, young people participants aged 10-18 years). Among those contacted, 1,400 (74.4%) guardians (in 93.1% of cases the biological mother) completed the interview, 982 (70.1%) by telephone and 418 (29.9%) face-to-face (See flow chart in S1 Fig.). There were no significant differences in persistence of psychopathology or impact of psychopathology on adolescents’ lives among respondents versus non-respondents.

Results

Follow-up query to comment 14: It was helpful to see the description of the results in text. A few comments on the revised text: In line 253, the value in the text doesn't match with the value given in the table. If the rounding up was done, the number of decimal places should be consistent across the text. In line 262, there is no description of whether this service use is significantly different from other trajectories?

We appreciate your comment. We have edited the value in line 259 (10.6%) and we have included the description of whether the service use was significantly different by trajectories:

Unadjusted odds ratios of any service use among participants with persistent diagnosis were 7.14 (95%CI=4.50-11.58, p<0.001) compared with participants with no diagnosis, OR=1.72 (95%CI=1.02-2.91, p=0.043) compared with incident and OR=2.62 (95%CI=1.52-4.49, p<0.001) with remittent diagnosis. (Lines 267-270).

Follow-up query to comment 16: Again, it was helpful to see the description of the results in text. A few comments on the revised text: In line 276, there should be a full stop before starting the following sentence. Also, it may be good to start as "within the health sector, the outpatient…", so the reader is clear that now you are looking at the frequency of usage within each sector.

Thank you very much for your suggestion, we have corrected this as requested. (Line 284)

In line 289, it might be good to specify the mean visit per person, so it's clear to the reader that the mean refers to the mean visit per person.

Thank you very much for your suggestion, we have corrected this as requested. (Line 297)

The source of data in lines 334-337 is not provided.

We appreciate your comment. Due to lines 334-337 are part of the first paragraph of the discussion, we wonder whether you are referring to lines 324-327. If so, we have edited this sentence as follows:

When looking at predictors of costs according to sector (Table 4), disorder impact was associated with greater health sector service use (predicted mean cost by each impact score= 66.26 USD, p=0.019). (Lines 343-345)

There are a lot of supplementary data files, but except for two supplementary files, others were not referenced or explained in the results section. It would be helpful to understand the reason for including all this analysis but not using them in results.

Supplementary analyses were explained in the methods section. Following your suggestion, we have included the description of supplementary tables in the results sections as follows:

Regarding to service use, lines 312-326:

Having an incident, remittent or persistent psychiatric disorder, as well as the higher impact of behavioural and emotional difficulties on the adolescents’ lives and lower parental stigma, all predicted higher odds of any 12-month service use in unadjusted analyses (S2 Table), in models adjusted by sociodemographic characteristics (S4-S6 Tables) and multivariable analyses (Table 3). Service contacts in the health sector were also predicted by the same factors. Service use in the educational sector was predicted by diagnosis trajectory, impact and lower stigma in the unadjusted analyses (S2 Table), and in the models adjusted by sociodemographic characteristics (S4-S6 Tables). However, disorder persistence did not remain significant in multivariable analyses, where impact, lower parental stigma and low SEG showed significant association with education service use (Table 3). Although persistence of the disorder and higher impact of behavioural and emotional difficulties on the adolescents’ lives were associated with social care and criminal justice service use in the unadjusted analyses (S2 Table), and in the models adjusted by sociodemographic characteristics (S4-S5 Table), there were no factors significantly associated with use of social care and criminal justice services in multivariable analyses (Table 3).

Additionally, to clarify that adjusted estimates are showed in Table 3, we have changed AOR (adjusted odds rations) instead OR (Line 328).

Regarding to services costs, lines 334-338:

When all three sectors were combined into a single total cost variable, greater impact and lower parental stigma were associated with higher costs in unadjusted analyses (S3 Table), in the models adjusted by sociodemographic characteristics (S8-S9 Tables) and multivariable analyses (Table 4). White ethnicity was also associated with higher costs in multivariable analyses (Table 4)….

Lines 345-350:

We did not find any significant association of psychiatric trajectories, impact of disorder or parental stigma with education sector’s costs (S3 Table, S7-S9 Tables, and Table 4). Although persistence of the disorder was associated with social care and criminal justice service’s costs in the unadjusted analyses (S3 Table), and in the models adjusted by sociodemographic characteristics (S7 Table), we did not find any significant factors associated with social care/criminal justice sectors’ costs in multivariable analyses (Table 4).

Discussion

The findings discussed in lines 342-346 don't match the result. The service use was different among those with different trajectories. However, the costs were not different among these groups. Does any of the existing analysis carried out by the author can explain why the difference in frequency of use didn't result in differences in cost between these groups.

Thanks for your comment. Apologise for the confusion. We have corrected that we were discussing in this part the results of service costs instead of service use, as follows:

Our analyses instead found that impact of the disorder on adolescent’s life was the most important clinical predictor and that this was what seemed to drive service costs rather than type or persistence of diagnosis. (Lines 366-368)

Regarding to your question about whether our analyses can explain why the difference in frequency of use did not result in differences in cost between these groups, service costs reflect the intensity of care (number of visits, as explained in the introduction) while service use reflect the probability of having any contact with the services. In this sense, our results suggest that the persistence of a diagnosis can increase the likelihood of service use but does not necessarily have an impact on the number of contacts per year. Unfortunately, as pointed out in the limitations of our study (lines 433-435), the scarce number of service users by specific services did not allow us to perform analyses to compare, for example, whether the trajectory it is associated to the selection of CAPS (high-costs services) instead primary care (low-costs services) services contacts. We have added the following sentence to the discussion:

This finding suggests that although the trajectory of the diagnosis is associated with a greater likelihood of having any contact with mental health-related services, this association did not translate to costs which also reflects the intensity and / or type of care and number of visits per year. Nevertheless, it is important to consider that we have estimated annual costs, and these do not necessarily reflect the cumulative economic costs of persistent cases across childhood and adolescence. (Lines 368-374).

Follow-up query to comment 18: The revision looks good. However, in line 347, the authors' assertion that these findings are novel seems misleading. As in quick succession, they have indicated that other studies have also found similar findings.

Thanks for your comment. We have edited this sentence as follows:

We also found that lower parental stigma was associated with greater service use and higher costs. (Line 371)

Additionally, as we are aware of a new paper (published on January 27th, 2022) that investigate the association between parental stigma and service use costs, we have edited the paragraph in lines (381-384):

We only found one study [20] exploring the impact of parental stigmatising attitudes toward mental illness on child service use costs. The cited study, conducted in the UK, with a smaller sample size, did not find an association between parental stigma and young people’s service use costs.

Follow-up query to comment 19: The discussion points on CAPS results in lines 380-385 are difficult to follow. There is no mention in results about the severity of disorders and types of service accessed. So understanding this link in discussion is difficult.

We appreciate your comment. We eliminated the sentence that connected severity of cases with CAPS’s costs and the sentence in the revised version appears as follows:

The high costs incurred by the mental health sector for the treatment of psychiatric disorders in CAPS may be because these services provide intensive outpatient treatments (reflected by the highest number of visits we found), which is costly compared with no-specialized services. (Lines 408-411)

Attachment

Submitted filename: Response to reviewers.docx

Decision Letter 2

Therese van Amelsvoort

25 May 2022

PONE-D-21-10663R2Utilisation and costs of mental health-related service use among adolescentsPLOS ONE

Dear Dr. Lacko,

Thank you for submitting your manuscript to PLOS ONE. There are still some minor questions remaining before we can accept your manuscript.

Please submit your revised manuscript by Jul 09 2022 11:59PM. If you will need more time than this to complete your revisions, please reply to this message or contact the journal office at plosone@plos.org. When you're ready to submit your revision, log on to https://www.editorialmanager.com/pone/ and select the 'Submissions Needing Revision' folder to locate your manuscript file.

Please include the following items when submitting your revised manuscript:

  • A rebuttal letter that responds to each point raised by the academic editor and reviewer(s). You should upload this letter as a separate file labeled 'Response to Reviewers'.

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If you would like to make changes to your financial disclosure, please include your updated statement in your cover letter. Guidelines for resubmitting your figure files are available below the reviewer comments at the end of this letter.

If applicable, we recommend that you deposit your laboratory protocols in protocols.io to enhance the reproducibility of your results. Protocols.io assigns your protocol its own identifier (DOI) so that it can be cited independently in the future. For instructions see: https://journals.plos.org/plosone/s/submission-guidelines#loc-laboratory-protocols. Additionally, PLOS ONE offers an option for publishing peer-reviewed Lab Protocol articles, which describe protocols hosted on protocols.io. Read more information on sharing protocols at https://plos.org/protocols?utm_medium=editorial-email&utm_source=authorletters&utm_campaign=protocols.

We look forward to receiving your revised manuscript.

Kind regards,

Therese van Amelsvoort

Academic Editor

PLOS ONE

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Please review your reference list to ensure that it is complete and correct. If you have cited papers that have been retracted, please include the rationale for doing so in the manuscript text, or remove these references and replace them with relevant current references. Any changes to the reference list should be mentioned in the rebuttal letter that accompanies your revised manuscript. If you need to cite a retracted article, indicate the article’s retracted status in the References list and also include a citation and full reference for the retraction notice.

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Reviewers' comments:

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Comments to the Author

1. If the authors have adequately addressed your comments raised in a previous round of review and you feel that this manuscript is now acceptable for publication, you may indicate that here to bypass the “Comments to the Author” section, enter your conflict of interest statement in the “Confidential to Editor” section, and submit your "Accept" recommendation.

Reviewer #1: All comments have been addressed

Reviewer #2: (No Response)

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2. Is the manuscript technically sound, and do the data support the conclusions?

The manuscript must describe a technically sound piece of scientific research with data that supports the conclusions. Experiments must have been conducted rigorously, with appropriate controls, replication, and sample sizes. The conclusions must be drawn appropriately based on the data presented.

Reviewer #1: (No Response)

Reviewer #2: Partly

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3. Has the statistical analysis been performed appropriately and rigorously?

Reviewer #1: (No Response)

Reviewer #2: Yes

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4. Have the authors made all data underlying the findings in their manuscript fully available?

The PLOS Data policy requires authors to make all data underlying the findings described in their manuscript fully available without restriction, with rare exception (please refer to the Data Availability Statement in the manuscript PDF file). The data should be provided as part of the manuscript or its supporting information, or deposited to a public repository. For example, in addition to summary statistics, the data points behind means, medians and variance measures should be available. If there are restrictions on publicly sharing data—e.g. participant privacy or use of data from a third party—those must be specified.

Reviewer #1: (No Response)

Reviewer #2: Yes

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5. Is the manuscript presented in an intelligible fashion and written in standard English?

PLOS ONE does not copyedit accepted manuscripts, so the language in submitted articles must be clear, correct, and unambiguous. Any typographical or grammatical errors should be corrected at revision, so please note any specific errors here.

Reviewer #1: (No Response)

Reviewer #2: Yes

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6. Review Comments to the Author

Please use the space provided to explain your answers to the questions above. You may also include additional comments for the author, including concerns about dual publication, research ethics, or publication ethics. (Please upload your review as an attachment if it exceeds 20,000 characters)

Reviewer #1: My comments were already addressed, to my satisfaction, in the previous version of the manuscript.

Reviewer #2: Thank you for addressing most of my comments. The manuscript is looking well. Here are a suggested few minor revisions:

In lines 59-61, it is still unclear whether these features increase the likelihood of service usage or the number of services used.

In lines 121-122, the authors have stated that 'due to an administrative error, we were only able to contact a subsample of those who participated at the first follow-up'. It will be good to have more details about this error in the study limitation and how this may have affected the validity of the results.

For lines 261-272, I'm really struggling to figure out where all these percentage values, OR, and RR are coming from, as they are not given in Table 1. If it's from some supplementary table, please cite it.

In lines 258 and 267, the value in the text doesn't match the value given in the table. If the rounding up was done, the number of decimal places should be consistent across the text

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Reviewer #1: No

Reviewer #2: Yes: KANIKA MALIK

[NOTE: If reviewer comments were submitted as an attachment file, they will be attached to this email and accessible via the submission site. Please log into your account, locate the manuscript record, and check for the action link "View Attachments". If this link does not appear, there are no attachment files.]

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PLoS One. 2022 Sep 9;17(9):e0273628. doi: 10.1371/journal.pone.0273628.r006

Author response to Decision Letter 2


17 Jun 2022

We appreciate the careful revision of our manuscript and the comments of the reviewers. We are pleased to be invited to submit the revised version of our paper to PLOS ONE.

Please find attached both an unmarked version of the revised manuscript and one version with changes marked in red. Our point-by-point responses to the reviewers’ comments (unquoted italics) and details of the changes we have performed to our revised manuscript are given below.

Reviewer #1: My comments were already addressed, to my satisfaction, in the previous version of the manuscript.

Many thanks for your revision and we are glad of having addressed your comments appropriately.

Reviewer #2: Thank you for addressing most of my comments. The manuscript is looking well. Here are a suggested few minor revisions:

In lines 59-61, it is still unclear whether these features increase the likelihood of service usage or the number of services used.

Thanks for your suggestion. We have edited this paragraph as follows:

Lines 59-62: Some studies from high–income countries about mental health-related service use among young people suggest that lower socioeconomic status, as well as clinical features (illness severity and impact of disorders) increase the likelihood of service usage in the health, special education, and social care sectors, while male gender and older age are associated with more criminal justice services contacts [11,13,14].

In lines 121-122, the authors have stated that 'due to an administrative error, we were only able to contact a subsample of those who participated at the first follow-up'. It will be good to have more details about this error in the study limitation and how this may have affected the validity of the results.

We appreciate your comment. We have provided more details about this error in lines 121-123 (Methods section):

Due to an administrative error, the service use questions were not included in the interview schedule for the first 129 participants (6%) of the BHRC first follow-up. Therefore, we were only able to contact a subsample of those who participated at the first follow-up (94%, n=1,881) to respond to a supplementary interview which included a comprehensive assessment of mental health-related service use (calendar year: 2014-2015, young people participants aged 10-18 years).

And we added in the Limitations subheading the following sentences:

Lines 440-442: Fifth, due to an administrative error we were unable to contact the first 6% of first follow-up BHRC participants. This could reduce our sample size, but as this was a random error, we do not believe that it affected the results.

For lines 261-272, I'm really struggling to figure out where all these percentage values, OR, and RR are coming from, as they are not given in Table 1. If it's from some supplementary table, please cite it.

We appreciate the comment of the reviewer.

To clarify the percentage of service utilization we edited the text as follows:

Lines 261-267: 23.3% (n= 260) of adolescents had a psychiatric disorder in the previous 12 months, of which (54.3%) 261 were incident and 149 (45.7%) persistent cases since baseline. 213 (15.2%) participants had remitted from a baseline psychiatric diagnosis. [We moved the next sentence here] 73 (22.4%) of those who presented with a psychiatric disorder (32 incident and 41 persistent) reported using some type of service for their mental health in the previous twelve months. The proportion of service use among those who presented a persistent psychiatric condition was 27.5%.

Regarding to the following text added in our second revision:

Lines 267-270: Unadjusted odds ratios of any service use among participants with persistent diagnosis were 7.22 [we amended these decimals] (95%CI=4.50-11.58, p<0.001) compared with participants with no diagnosis, OR=1.72 (95%CI=1.02-2.91, p=0.043) compared with incident and OR=2.62 (95%CI=1.52-4.49, p<0.001) with remittent diagnosis.

These results were added with the aim of addressing your comment on whether there were differences in service use between trajectories. In Supplementary Table 2 we presented these differences taking as reference ‘no diagnosis’, and the results presented in this text (Lines 267-270) would be taking as reference ‘persistent psychiatric conditions’. These results were not presented in a specific table.

Regarding to other statistics informed in the lines cited by the reviewer, Table 1 describes sociodemographic and clinical characteristics of participants, according to mental health conditions trajectories. We added only in the text (i.e. not in a specific table) the results of bivariate statistics for disorder impact (one of our main predictors) and type of broad diagnostic group by trajectories. To clarify this, we decided to only maintain the mean differences in disorder impact by trajectories and we removed the differences in terms of type of diagnostic group, as this was not the focus of our study. Changes were added as follows:

Lines 271-275: Table 1 present mean disorder impact by psychiatric trajectories. Unadjusted generalised regression models showed that persistent cases presented greater mean difference in disorder impact (SDQ scores) by 2.34, (95%CI=2.11-2.58, p<0.001) compared with no diagnosis, 1.14 (95% CI=0.85-1.42, p<0.001), compared with incident cases, and 1.84 (95% CI=1.56-2.12, p<0.001), compared with remittent cases.

In lines 258 and 267, the value in the text doesn't match the value given in the table. If the rounding up was done, the number of decimal places should be consistent across the text

Thanks for your suggestion, we amended these values as follows:

Line 260

14.51 years (s.d=1.98).

Line 267

27.5%.

And we also corrected the decimal places in the percentage of overall service use in Line 285 and Table 1

10.2%

Attachment

Submitted filename: Response to Reviewers.docx

Decision Letter 3

Dylan A Mordaunt

12 Aug 2022

Utilisation and costs of mental health-related service use among adolescents

PONE-D-21-10663R3

Dear Dr. Evans-Lacko,

We’re pleased to inform you that your manuscript has been judged scientifically suitable for publication and will be formally accepted for publication once it meets all outstanding technical requirements.

Within one week, you’ll receive an e-mail detailing the required amendments. When these have been addressed, you’ll receive a formal acceptance letter and your manuscript will be scheduled for publication.

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If your institution or institutions have a press office, please notify them about your upcoming paper to help maximize its impact. If they’ll be preparing press materials, please inform our press team as soon as possible -- no later than 48 hours after receiving the formal acceptance. Your manuscript will remain under strict press embargo until 2 pm Eastern Time on the date of publication. For more information, please contact onepress@plos.org.

Kind regards,

Dylan A Mordaunt, MD, MPH, FRACP

Academic Editor

PLOS ONE

Additional Editor Comments (optional):

Thank you for your resubmission. This now meets the criteria for publication.

Reviewers' comments:

Reviewer's Responses to Questions

Comments to the Author

1. If the authors have adequately addressed your comments raised in a previous round of review and you feel that this manuscript is now acceptable for publication, you may indicate that here to bypass the “Comments to the Author” section, enter your conflict of interest statement in the “Confidential to Editor” section, and submit your "Accept" recommendation.

Reviewer #1: All comments have been addressed

**********

2. Is the manuscript technically sound, and do the data support the conclusions?

The manuscript must describe a technically sound piece of scientific research with data that supports the conclusions. Experiments must have been conducted rigorously, with appropriate controls, replication, and sample sizes. The conclusions must be drawn appropriately based on the data presented.

Reviewer #1: (No Response)

**********

3. Has the statistical analysis been performed appropriately and rigorously?

Reviewer #1: (No Response)

**********

4. Have the authors made all data underlying the findings in their manuscript fully available?

The PLOS Data policy requires authors to make all data underlying the findings described in their manuscript fully available without restriction, with rare exception (please refer to the Data Availability Statement in the manuscript PDF file). The data should be provided as part of the manuscript or its supporting information, or deposited to a public repository. For example, in addition to summary statistics, the data points behind means, medians and variance measures should be available. If there are restrictions on publicly sharing data—e.g. participant privacy or use of data from a third party—those must be specified.

Reviewer #1: (No Response)

**********

5. Is the manuscript presented in an intelligible fashion and written in standard English?

PLOS ONE does not copyedit accepted manuscripts, so the language in submitted articles must be clear, correct, and unambiguous. Any typographical or grammatical errors should be corrected at revision, so please note any specific errors here.

Reviewer #1: (No Response)

**********

6. Review Comments to the Author

Please use the space provided to explain your answers to the questions above. You may also include additional comments for the author, including concerns about dual publication, research ethics, or publication ethics. (Please upload your review as an attachment if it exceeds 20,000 characters)

Reviewer #1: My comments were already addressed, to my satisfaction, in the previous version of the manuscript.

**********

7. PLOS authors have the option to publish the peer review history of their article (what does this mean?). If published, this will include your full peer review and any attached files.

If you choose “no”, your identity will remain anonymous but your review may still be made public.

Do you want your identity to be public for this peer review? For information about this choice, including consent withdrawal, please see our Privacy Policy.

Reviewer #1: No

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Acceptance letter

Dylan A Mordaunt

1 Sep 2022

PONE-D-21-10663R3

Utilisation and costs of mental health-related service use among adolescents

Dear Dr. Evans-Lacko:

I'm pleased to inform you that your manuscript has been deemed suitable for publication in PLOS ONE. Congratulations! Your manuscript is now with our production department.

If your institution or institutions have a press office, please let them know about your upcoming paper now to help maximize its impact. If they'll be preparing press materials, please inform our press team within the next 48 hours. Your manuscript will remain under strict press embargo until 2 pm Eastern Time on the date of publication. For more information please contact onepress@plos.org.

If we can help with anything else, please email us at plosone@plos.org.

Thank you for submitting your work to PLOS ONE and supporting open access.

Kind regards,

PLOS ONE Editorial Office Staff

on behalf of

Associate Professor Dylan A Mordaunt

Academic Editor

PLOS ONE

Associated Data

    This section collects any data citations, data availability statements, or supplementary materials included in this article.

    Supplementary Materials

    S1 Fig. Flow chart of Brazilian High-Risk Cohort participants included in the mental-health related service use study.

    (PDF)

    S1 Table. Unit costs of health, educational, social care and criminal justice related services.

    (PDF)

    S2 Table. Bivariate analysis: Predictors of 12-month mental health service utilisation.

    (PDF)

    S3 Table. Bivariate analysis: Predictors of cost of mental health service use in the last 12 months.

    (PDF)

    S4 Table. Logistic regression models: 12-month mental health service utilisation predicted by psychiatric diagnosis trajectories.

    (PDF)

    S5 Table. Logistic regression models: 12-month mental health service utilisation predicted by impact of behavioural and emotional difficulties on child’ life.

    (PDF)

    S6 Table. Logistic regression models: 12-month mental health service utilisation predicted by parental stigma.

    (PDF)

    S7 Table. Generalised linear models: Cost of 12-month mental health service utilization predicted by psychiatric diagnosis trajectories.

    (PDF)

    S8 Table. Generalised linear models: Cost of 12-month mental health service utilisation predicted by impact of behavioural and emotional difficulties on child’ life.

    (PDF)

    S9 Table. Generalised linear models: Cost of 12-month mental health service utilisation predicted by parental stigma.

    (PDF)

    S10 Table. Generalised linear models: Cost of 12-month mental health service utilisation predicted by broad diagnosis categories.

    (PDF)

    Attachment

    Submitted filename: Response to Reviewers.docx

    Attachment

    Submitted filename: PONE-D-21-10663_R1_WC.pdf

    Attachment

    Submitted filename: Response to reviewers.docx

    Attachment

    Submitted filename: Response to Reviewers.docx

    Data Availability Statement

    Access to data and Data sharing: CZ have full access to all the data used in the study and takes responsibility for the integrity of the data and the accuracy of the data analysis. Data were provided by the Brazilian High-Risk Cohort study and are available upon request in the Open Science Framework public repository (https://osf.io/ktz5h/).


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