Rehabilitation status of children with cerebral palsy in Bangladesh: Findings from the Bangladesh Cerebral Palsy Register

Mahmudul Hassan Al Imam; Israt Jahan; Manik Chandra Das; Mohammad Muhit; Hayley Smithers-Sheedy; Sarah McIntyre; Nadia Badawi; Gulam Khandaker

doi:10.1371/journal.pone.0250640

. 2021 May 3;16(5):e0250640. doi: 10.1371/journal.pone.0250640

Rehabilitation status of children with cerebral palsy in Bangladesh: Findings from the Bangladesh Cerebral Palsy Register

Mahmudul Hassan Al Imam ^1,^2,³, Israt Jahan ^1,^2,³, Manik Chandra Das ^1,², Mohammad Muhit ^1,², Hayley Smithers-Sheedy ⁴, Sarah McIntyre ⁴, Nadia Badawi ^4,⁵, Gulam Khandaker ^1,^2,^6,^7,^*

Editor: Enamul Kabir⁸

PMCID: PMC8092763 PMID: 33939721

Abstract

Objective

The objective of this study was to assess the rehabilitation status and factors associated with rehabilitation service utilisation among children with cerebral palsy (CP) in Bangladesh.

Materials and methods

This is a population-based surveillance study conducted among children with CP registered in the Bangladesh CP Register (BCPR), the first population-based register of children with CP aged <18 years (y) in Bangladesh. Children with CP were identified from the community using the key informant method and underwent a detailed neurodevelopmental assessment. Socio-demographic, clinical and rehabilitation status were documented. Unadjusted and adjusted analyses with a 95% confidence interval (CI) were used to identify potential predictors of rehabilitation service uptake.

Results

Between January 2015 and December 2019, 2852 children with CP were registered in the BCPR (mean (standard deviation, SD) age: 7 y 8 months (mo) (4 y 7 mo), 38.5% female). Of these, 50.2% had received rehabilitation services; physiotherapy was the most common type of service (90.0%). The mean (SD) age at commencement of rehabilitation services was 3 y 10 mo (3 y 1 mo). The odds of not receiving rehabilitation was significantly higher among female children (adjusted odds ratio (aOR) 1.3 [95% CI: 1.0–1.7], children whose mothers were illiterate and primary level completed (aOR 2.1 [95% CI: 1.4–3.1] and aOR 1.5 [95% CI: 1.1–2.1], respectively), fathers were illiterate (aOR 1.9 [95% CI: 1.3–2.8]), had a monthly family income ~US$ 59–118 (aOR: 1.8 [95% CI: 1.2–2.6]), had hearing impairment (aOR: 2.3 [95% CI: 1.5–3.5]) and motor severity (i.e. Gross Motor Function Classification System level III (aOR: 0.6 [95% CI: 0.3–0.9]) and level V (aOR: 0.4 [95% CI: 0.2–0.7])).

Conclusions

Rehabilitation status was poor among the majority of the children with CP in the BCPR cohort, limiting their opportunities for functional improvement. A community-based rehabilitation model focusing on socio-demographic and clinical characteristics should be a public health priority in Bangladesh.

Introduction

Childhood disability is a global public health concern due to its lifelong impact on physical and psychological wellbeing. An estimated 80% of childhood disability occurs in low- and middle-income countries (LMICs) [1]. Despite this high burden, there is limited information on access to rehabilitation services in LMICs [2]. The 2030 Agenda for Sustainable Development Goals warrants that children with disabilities should enjoy equal access to health care and rehabilitation (Goal #3 Good health and wellbeing) regardless of their abilities and socio-economic status (Goal #10 Reduce inequality).

Cerebral palsy (CP) is one of the leading causes of childhood disability, with an estimated 50 million people living with CP worldwide [3,4]. CP is a clinical description for non-progressive motor disorders caused by injury to the developing brain [5]. The burden of CP is estimated to be substantially higher in LMICs compared to high-income countries (HICs) [6,7].

Children with CP require support from a multidisciplinary team of medical and rehabilitation professionals including physiotherapists, occupational therapists and speech and language therapists to improve function, prevent secondary complications and enhance autonomy [8]. However, such services are not always available for children with CP, particularly in LMICs [9,10]. In Bangladesh, the population-based prevalence of CP was estimated to be 3.4 (95% CI 3.2–3.7) per 1000 children, which approximates to ~234,000 children with CP in a country of 166 million people [7]. However, this is likely to be an underestimation due to survival bias.

The utilisation of rehabilitation services for children with CP is multidimensional and is affected by many social, economic and ecological factors [11]. Younger age [12–19], male gender [20], high family income [7,10,16,21–24], parental education [7,22] and severe motor impairment [17,25–27] are positively associated with rehabilitation service uptake. Conversely, lack of access to information [10,24], lack of transportation support [7,22,24,25] and parents’ lack of awareness [7,22,24,28] have been reported as barriers to rehabilitation service utilisation. However, a majority of these studies have been conducted in HICs [14,15,17–19,27,28]. Studies completed in LMICs to date have largely been conducted in hospital settings [13,20,22] and have focused on a particular service (i.e. physiotherapy) [22,24]. Furthermore, the limited service availability and shortage of rehabilitation services in LMICs make the situation more complicated.

Population-based data in this regard is limited in LMICs. Such data are essential to identify potential scope for intervention, ensure optimal use of inadequate available resources and maximise the service coverage for children with disabilities (e.g. CP) in the resource-constrained settings of LMICs like Bangladesh. Therefore, this study aimed to assess the rehabilitation status and the factors associated with rehabilitation service uptake among children with CP in Bangladesh.

Materials and methods

We established the first population-based surveillance of children with CP i.e. Bangladesh CP Register (BCPR) in rural Bangladesh in 2015. The BCPR is an ongoing surveillance that studies epidemiology, rehabilitation and intervention strategies to improve functional outcomes and limit associated impairments among children with CP in Bangladesh [29]. Initially, the surveillance activities (i.e. BCPR) were confined in one subdistrict (Shahjadpur, ~325 square kilometres, ~123,576 households) which represents rural and semi-urban Bangladesh (where the majority of the population (76.7%) lives [30]) in terms of demographic and other indicators (e.g. birth rate, immunisation rate, perinatal mortality rate, literacy rate). To date, this population-based surveillance has been maintained with high case ascertainment in Shahjadpur. Additionally, between 2015 and 2019 the BCPR has been scaled up to 17 other subdistricts (Σ4,338 square kilometres, ~1,304,960 households) following opportunistic recruitment at the community level (Fig 1).

Fig 1 — Circle diameters are proportional to the number of children with CP recruited in the study. (This map was produced by the authors using ArcGIS Desktop 10.8 software).

Study participants and data collection method

As part of the ongoing surveillance (i.e. BCPR), children with suspected CP in a community were identified using the key informant method (KIM). The KIM is a validated method where local volunteers (e.g. religious leaders, teachers, community health workers etc.) are trained as key informants (KIs) to identify children with disabilities in their communities [31]. The KIs were identified (approximately 1 KI per village) by the Community Mobilisers (CMs—paid project staff) and received a day-long training on the identification of children with suspected CP, disability sensitisation, advocacy for a disability inclusive society using flip charts, group work and role play. Following the training, the KIs were provided 4–6 weeks to identify and enlist children with suspected CP and share their contact details with CMs to bring those children and their primary caregivers to the nearest medical assessment camps for a confirmed diagnosis, detailed neurodevelopmental assessments and registration in the BCPR. The clinical definition of CP adopted from the Surveillance of CP in Europe (SCPE) [32] and the Australian CP Register (ACPR) [6] were strictly followed during case ascertainment. The details of case identification and recruitment into the BCPR have been described in our previous publication [7].

Data were collected using a standard case-record form (adapted from the ACPR) through interviews with the primary caregivers, as well as a clinical assessment and a review of medical records (if available). The detailed data collection method and the variables included in the BCPR are available in our previous publication [7]. In the current analyses, we used the following variables: (i) socio-demographic characteristics (e.g. age, gender, educational level of parents, monthly family income); (ii) clinical characteristics (e.g. Gross Motor Function Classification System (GMFCS) level, Manual Ability Classification System (MACS) level, predominant CP motor type, CP topography, associated impairments) and (iii) information on rehabilitation (i.e. whether the child ever received rehabilitation, the type and location/source of services received, age of commencement of rehabilitation). Here, the main outcome variable was the rehabilitation status (i.e. whether he/she ever received rehabilitation) of a child with CP registered in the BCPR, which is a binary variable with ‘yes’ and ‘no’ as responses. Information on rehabilitation was collected from primary caregivers of children with CP by a physiotherapist at the medical assessment camps in the context of BCPR recruitment. To document the type of rehabilitation services received, multiple responses were allowed and, therefore, the numbers presented for this variable are not mutually exclusive. Additionally, the medical records available from primary caregivers were reviewed for any documentation of rehabilitation services.

Data management and analysis

Following collection, the data were entered electronically into the online password-protected BCPR data repository (http://bangladesh.cpregister.com/), access to which was limited to named investigators only. A dedicated data management team located at CSF Global in Dhaka, Bangladesh, with support from investigators conducted the data management and analyses.

To maintain the quality of the data, we used a double data entry method. Subsequently, data-entry error checks were performed by running frequencies of all variables to identify any outliers. In case of any missing data or incorrect/suspicious information, the BCPR case record forms (i.e. source documents) were reviewed. If the information was not found in the BCPR form, it was relayed to the field team and where possible the participants were contacted to gather the missing information. Continuous variables were collected as exact values and later recoded and categorised into groups (e.g. age was categorised as follows: 0–4 years, 5–9 years, 10–14 years and 15–18 years). Similarly, the monthly family income data were converted to United States Dollar (US$) (considering 1 US$ = 84.43 Bangladeshi taka (BDT)) and categorised into four family-income groups (i.e. BDT 500–4999 (US$ Σ6–59), BDT 5000–9999 (US$ Σ59–118), BDT 10,000–14,999 (US$ Σ118–178) and BDT 15,000 and above (US$ Σ178 and above). Comparison between the BCPR cohort and the general population was performed using the 2014 Bangladesh Demographic and Health Survey (BDHS) [33] and Household Income and Expenditure Survey [34] data from 2016. The poverty level among the families in the BCPR cohort was estimated using the national poverty lines (at the divisional level) as a cut-off. The proportion of families living below the poverty lines was then compared with the general population of the respective divisions as reported in the 2016 Household Income Expenditure Survey (HIES) in Bangladesh [34]. Bivariate analyses were completed to assess the impact of socio-demographic and clinical factors on rehabilitation status. For regression models, ‘Not receiving rehabilitation’ was considered as the main outcome of interest. Factors that were found to be statistically significant in unadjusted logistic regression were fitted into an adjusted logistic regression model. Odds ratios with 95% confidence intervals (CI) were reported. A p value <0.05 was considered significant. All data were analysed using the Statistical Package for the Social Sciences (SPSS) software, version 26 (IBM, Armonk, NY, USA).

Ethical considerations

Ethical approval for the BCPR study was obtained from the Cerebral Palsy Alliance Human Research Ethics Committee (EC00402; ref no. 2015-03-02) in Australia, the Asian Institute of Disability and Development Human Research Ethics Committee (southasia-irb-2014-l-01), and the Bangladesh Medical Research Council National Research Ethics Committee (BMRC/NREC/2013-2016/1267) in Bangladesh. Written informed consent was given by the parents or primary caregivers of all study participants prior to data collection and registration in the BCPR.

Results

Between January 2015 and December 2019, 2852 children with CP were registered in the surveillance study (i.e. BCPR). The mean age at assessment was 7 years (y) and 8 months (mo) (standard deviation (SD) 4 y and 7 mo; median 7 y 1 mo; interquartile range (IQR) 3 y 10 mo–11 y 4 mo); 38.5% (n = 1097/2852) female.

Socio-demographic characteristics

The age and sex distributions of participating children were significantly different than the general population (children aged less than 10 y: 68.5% vs 49.0%; p<0.001, and male–female ratio 1.6:1 vs 1.1:1; p<0.001 in the BCPR vs the general population, respectively). Compared to the national data, fewer mothers and fathers of children in the BCPR cohort were educated (illiterate mothers: 30.0% vs 26.7%, and illiterate fathers: 39.2% vs 22.9%; p<0.001 in the BCPR vs the general population, respectively. Overall, 73.3% (n = 2074) children in the BCPR were from families living below the national poverty line, compared with 24.0% in the general population (p<0.001). The median monthly income of families was BDT 8000 (US$ ~95; IQR BDT 6000–10,000 (US$ ~71–118), mean BDT 9417 (US$ ~112) and SD BDT 7916.5 (US$ ~94)) (Table 1).

Table 1. Socio-demographic characteristics of children with CP.

Characteristics	BCPR n (%)	General population %	p value
Age group in years (n = 2799)^a
0–4	947 (33.8)	23.5	<0.001^e
5–9	972 (34.7)	25.5
10–14	621 (22.2)	27.1
15–18	259 (9.3)	23.9^b
Sex (n = 2852)
Male	1755 (61.5)	51.3^b	<0.001^e
Female	1097 (38.5)	48.7	<0.001^e
Mother’s education (n = 2845)^a
No education	854 (30.0)	26.7	<0.001^e
Primary	1134 (39.9)	35.3
Secondary and above	857 (30.1)	38.0^b
Father’s education (n = 2827)^a
No education	1108 (39.2)	22.9	<0.001^e
Primary	881 (31.2)	37.6
Secondary and above	838 (29.6)	39.5^b
Monthly family income, BDT (US$), (n = 2828)^a^,^c
BDT 500–4999 (US$ Σ6–59)	210 (7.4)	N/A	N/A
BDT 5000–9999 (US$ Σ59-–18)	1704 (60.3)	N/A	N/A
BDT 10,000–14,999 (US$ Σ118–178)	510 (18.0)	N/A	N/A
BDT 15,000 and above (US$ Σ178 and above	404 (14.3)	N/A	N/A
Families below the national poverty line (minimum–maximum according to divisions)	2074 (73.3) [22 (50.0) –1984 (74.2)]	24.0 (16.0–32.8)^d	<0.001^f

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^aMissing data exists (Age group in years = 53; Mothers’ education = 7; Fathers’ education = 25; Monthly family income = 24).

^bData from the 2014 Bangladesh Demographic and Health Survey (BDHS), and 23.9% for the general population refers to the 15–19 years age group [33].

^cUS$ 1 = BDT 84.43.

^dData from the 2016 Household Income and Expenditure Survey [34].

^eChi-squared test.

^fBinomial test.

Rehabilitation status

Almost half of the study participants (49.8%, n = 1411/2836) had never received any rehabilitation services. Among children who had received rehabilitation services, 90.0% (n = 1264/1404) received physiotherapy. Of children with a GMFCS level III–V (73.7%, n = 2090/2836), only 4.7% (n = 98/2090) received any assistive devices. The mean age for commencing rehabilitation services was 3 y 10 mo (SD 3 y 1 mo; median 3 y 0 mo; IQR 1 y 6 mo–5 y 0 mo). Nearly one-third of children (30.4%, n = 415/1365) first received rehabilitation services at or over 5 y of age. The most common rehabilitation service providers were centres run by non-governmental organisations (45.1%, n = 626/1387) (Table 2).

Table 2. Rehabilitation status of children with CP in the BCPR cohort.

Variable Name	BCPR N = 2852 (%)
Rehabilitation service received (n = 2836)^a
No	1411 (49.8)
Yes	1425 (50.2)
Type of rehabilitation service received (n = 1404)^a^,^b
Physiotherapy	1264 (90.0)
Advice	156 (11.1)
Assistive device	124 (8.8)
Primary location of rehabilitation service (n = 1387)^a
Non-governmental organisation centre	626 (45.1)
Hospital	443 (31.9)
Home-based	153 (11.0)
Private clinic	141 (10.2)
Special school	24 (1.7)
Age (y) at first commencement of rehabilitation service (n = 1365)^a
<5	950 (69.6)
5–10	324 (23.7)
10 and above	91 (6.7)

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^aMissing data exists (Rehabilitation service received = 16; Type of rehabilitation service received = 21; Primary location of rehabilitation service = 38; Age at first commencement of rehabilitation = 60).

^bNot mutually exclusive.

Factors influencing receipt of rehabilitation services among children with CP

Age and sex of children with CP

Children in the BCPR cohort aged 10–14 y and 15–18 y had 1.3 times (95% confidence interval (CI) 1.1–1.7) and 1.6 times (95% CI 1.2–2.2) lower likelihood of receiving rehabilitation services compared with children aged 0–4 y, respectively. Female children were 1.2 times (95% CI 1.0–1.4) less likely to receive rehabilitation services compared to male children (Table 3).

Table 3. Socio-demographic factors related to the receipt of rehabilitation for children with CP in the BCPR cohort.

Characteristics n = 2852	Total n (%)	Ever received rehabilitation^a			Unadjusted OR for not receiving rehabilitation (CI)	p value^d
Characteristics n = 2852	Total n (%)	No n (%)	Yes n (%)	p value	Unadjusted OR for not receiving rehabilitation (CI)	p value^d
Age (n = 2799)^a
0–4	947 (33.8)	432 (46.0)	507 (54.0)	0.001^b	Ref
5–9	972 (34.7)	466 (48.1)	502 (51.9)		1.1 (0.9 1.3)	0.351
10–14	621 (22.2)	331 (53.5)	288 (46.5)		1.3 (1.1 1.7)	0.004
15–18	259 (9.3)	150 (58.4)	107 (41.6)		1.6 (1.2 2.2)	<0.001
Sex (n = 2852)
Male	1755 (61.5)	837 (47.9)	910 (52.1)	0.013 ^b	Ref
Female	1097 (38.5)	574 (52.7)	515 (47.3)	0.013 ^b	1.2 (1.0 1.4)	0.013
Mothers’ education (n = 2845)^a
No education	854 (30.0)	534 (62.8)	316 (37.2)	<0.001^b	3.4 (2.8 4.2)	<0.001
Primary completed	1134 (39.9)	592 (52.6)	533 (47.4)		2.3 (1.9 2.7)	<0.001
Secondary and above	857 (30.1)	282 (33.0)	572 (67.0)		Ref
Fathers’ education (n = 2827)^a
No education	1108 (39.2)	673 (61.0)	430 (39.0)	<0.001^b	2.8 (2.3 3.3)	<0.001
Primary completed	881 (31.2)	428 (48.9)	448 (51.1)		1.7 (1.4 2.0)	<0.001
Secondary and above	838 (29.6)	301 (36.2)	531 (63.8)		Ref
Monthly family income (n = 2828)^a
BDT 500–4999 (US$ Σ6–59)	210 (7.4)	104 (50.2)	103 (49.8)	<0.001^b	1.9 (1.4 2.7)	<0.001
BDT 5000–9999 (US$ Σ59–118)	1704 (60.3)	936 (55.3)	758 (44.7)		2.4 (1.9 3.0)	<0.001
BDT 10,000–14,999 (US$ Σ118–178)	510 (18.0)	221 (43.6)	286 (56.4)		1.5 (1.1 1.9)	0.005
BDT 15,000 and above (US$ Σ178 and above	404 (14.3)	139 (34.4)	265 (65.6)		Ref
Median (IQR) monthly family income (n = 2812)^a	BDT 8000 (6000–10,000)/US$ Σ95 (71–118)	BDT 7000 (6000–10,000)/US$ Σ83 (71–118)	BDT 8000 (6000–11,375)/US$ Σ95 (71–135)	<0.001^c

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^aMissing data exists (Ever received rehabilitation = 16; Age = 53; Mothers’ education = 7; Fathers’ education = 25; Monthly family income = 24).

^bChi-squared test.

^cMann–Whitney U test.

^dLogistic regression.

Education of parents

Parental education was significantly related to rehabilitation service uptake among children with CP in our cohort (p<0.001). Of the mothers and fathers of children with CP who had received rehabilitation services, 77.8% (n = 1105/1421) and 69.5% (n = 979/1409) had completed primary orsecondary education. Children of illiterate mothers had a 3.4 times (95% CI 2.8–4.2) lower chance of receiving rehabilitation services. A similar observation was reported for the father’s education (Table 3).

Monthly family income

A significant negative association between monthly family income and rehabilitation service uptake was observed in our cohort. The median (IQR) monthly family income of children who had received and had never received rehabilitation services was BDT 8000 (6000–11,375) [US$ Σ95 (Σ71–135)] and BDT 7000 (6000–10,000) [US$ Σ83 (Σ71–118)], respectively (p<0.001). Children from families with a monthly income of BDT 5000–9999 (US$ Σ59–118) were 2.4 (95% CI 1.9–3.0) times less likely to receive rehabilitation services than children with a monthly family income of BDT 15,000 (US$ Σ178) and above (Table 3).

Predominant motor type and topography of CP

Rehabilitation service utilisation was highest among children with dyskinesia and lowest among children with ataxia (52.3% (n = 92/176) vs 42.0% (n = 37/88), respectively). Compared to children with spastic CP, ataxic children were 1.4 times (95% CI 0.9–2.2) less likely to receive rehabilitation services. Furthermore, among children with spastic CP, tri/quadriplegic children had a 70% higher chance (95% CI 0.6–0.8) of receiving rehabilitation services than children with mono/hemiplegia (Table 4).

Table 4. Clinical factors related to the receipt of rehabilitation for children with CP in the BCPR cohort.

Characteristics	Total n (%)	Ever received rehabilitation^a			Unadjusted OR for not receving rehabilitation (CI)	p value^e
Characteristics	Total n (%)	No n (%)	Yes n (%)	p value	Unadjusted OR for not receving rehabilitation (CI)	p value^e
Predominant motor type of CP (n = 2852)
Spastic	2293 (80.4)	1129 (49.4)	1155 (50.6)	0.401^c	Ref
Dyskinetic	179 (6.3)	84 (47.7)	92 (52.3)		0.9 (0.7 1.3)	0.663
Ataxic	91 (3.2)	51 (58.0)	37 (42.0)		1.4 (0.9 2.2)	0.118
Hypotonic	289 (10.1)	147 (51.0)	141 (49.0)		1.1 (0.8 1.4)	0.606
CP topography (n = 2293)
Monoplegia and hemiplegia	638 (27.8)	350 (54.9)	287 (45.1)	<0.001^c	Ref
Diplegia	410 (17.9)	219 (53.4)	191 (46.6)		0.9 (0.7 1.2)	0.628
Triplegia and quadriplegia	1245 (54.3)	560 (45.3)	677 (54.7)		0.7 (0.6 0.8)	<0.001
GMFCS level (n = 2836)^a
I	252 (8.9)	151 (60.2)	100 (39.8)	<0.001^c	Ref
II	494 (17.4)	269 (54.9)	221 (45.1)		0.8 (0.6 1.1)	0.172
III	599 (21.1)	298 (50.0)	298 (50.0)		0.7 (0.5 0.9)	0.007
IV	492 (17.3)	258 (52.5)	233 (47.5)		0.7 (0.5 1.0)	0.049
V	999 (35.2)	432 (43.5)	561 (56.5)		0.5 (0.4 0.7)	<0.001
MACS level (n = 2220)^a^,^b
I	305 (13.7)	156 (51.3)	148 (48.7)	0.001^c	Ref
II	391 (17.6)	190 (49.0)	198 (51.0)		0.9 (0.7 1.2)	0.540
III	422 (19.0)	193 (46.1)	226 (53.9)		0.8 (0.6 1.1)	0.163
IV	423 (19.1)	199 (47.3)	222 (52.7)		0.9 (0.6 1.1)	0.282
V	679 (30.6)	262 (38.8)	413 (61.2)		0.6 (0.5 0.8)	<0.001
Type of associated impairment
Epilepsy (n = 2835)^a^,^d	897 (31.6)	428 (48.0)	464 (52.0)	0.197^c	0.9 (0.8 1.1)	0.197
Intellectual (n = 1944)^a^,^d	1074 (55.2)	570 (53.4)	497 (46.6)	0.020^c	1.2 (1.0 1.5)	0.020
Visual (n = 2813)^a^,^d	462 (16.4)	257 (56.1)	201 (43.9)	0.004^c	1.3 (1.1 1.6)	0.004
Hearing (n = 2835)^a^,^d	580 (20.5)	364 (63.2)	212 (36.8)	<0.001^c	2.0 (1.6 2.4)	<0.001
Speech (n = 2834)^a^,^d	2132 (75.2)	1054 (49.7)	1066 (50.3)	0.744^c	1.0 (0.8 1.2)	0.744
Number of associated impairments (n = 1889)^a
None	412 (21.8)	212 (51.7)	198 (48.3)	0.001^c	Ref
1–2 impairments	870 (46.1)	406 (46.9)	460 (53.1)		0.8 (0.7 1.0)	0.107
3–5 impairments	607 (32.1)	349 (57.9)	254 (42.1)		1.3 (1.0 1.7)	0.053

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^aMissing data exists (Ever received rehabilitation = 16; GMFCS level = 16; MACS level = 21; Epilepsy = 17; Intellectual impairment = 908; Visual impairment = 39; Hearing impairment = 17; Speech impairment = 18; Number of associated impairments = 963).

^bMACS was assessed among children aged four years of age or over.

^cChi-squared test.

^dReference category: No impairment.

^eLogistic regression.

GMFCS and MACS level

GMFCS level III–V and MACS level III–V were significantly overrepresented among children who had received rehabilitation services (77.3%, n = 1092/1413, p<0.001, and 71.3%, n = 861/1207, p = 0.001, respectively). Children with GMFCS level V were 50% more likely (95% CI 0.4–0.7) to receive rehabilitation services compared to children with GMFCS level I (p<0.001). A similar association was observed between rehabilitation service utilisation and the MACS level of children in the BCPR cohort (Table 4).

Associated impairments

Children with 3–5 associated impairments had a lower likelihood of receiving rehabilitation services than children with 1–2 impairments (odds ratio (OR): 1.3 (1.0–1.7) vs OR 0.8 (0.7–1.0), respectively). Furthermore, rehabilitation service utilisation was significantly lower among children with intellectual impairment (46.6%, n = 497/1067; p = 0.020), visual impairment (43.9%, n = 201/458; p = 0.004) and hearing impairment (36.8%, n = 212/576; p<0.001) (Table 4).

Independent predictors of not receiving rehabilitation services among children with CP in the BCPR cohort

Child’s gender, maternal and paternal education, monthly family income, GMFCS level, and the presence of hearing impairment were found to be significantly associated with rehabilitation service utilisation for children with CP registered in the BCPR when adjusted for other socio-demographic and clinical factors. The adjusted odds ratios (aORs) for not receiving rehabilitation were 1.3 (95% CI 1.0–1.7) for female children, 2.1 (95% CI 1.4–3.1) and 1.5 (95% CI 1.1–2.1) among children whose mothers were illiterate and primary completed, respectively, 1.9 (95% CI 1.3–2.8) among children whose fathers were illiterate, 1.8 (95% CI 1.2–2.6) among children with a monthly family income of BDT 5000–9999 (US$ 59–118), 0.6 (95% CI 0.3–0.9) and 0.4 (95% CI 0.2–0.7) among children with GMFCS level III and level V, respectively and 2.3 (95% CI 1.5–3.5) among children with hearing impairment (Table 5).

Table 5. Predictors of not receiving rehabilitation service for children with CP in the BCPR cohort.

Characteristics	Not receiving rehabilitation^a
Characteristics	Adjusted OR (CI)	p value
Age
0–4	Ref
5–9	0.9 (0.6 1.3)	0.521
10–14	1.1 (0.8 1.6)	0.546
15–18	1.4 (0.9 2.2)	0.191
Sex
Male	Ref
Female	1.3 (1.0 1.7)	0.034
Mothers’ education
No education	2.1 (1.4 3.1)	<0.001
Primary completed	1.5 (1.1 2.1)	0.013
Higher than primary	Ref
Fathers’ education
No education	1.9 (1.3 2.8)	0.001
Primary completed	1.2 (0.9 1.7)	0.263
Higher than primary	Ref
Family income
BDT 500–4999 (US$ Σ6–59)	1.6 (0.9 2.8)	0.095
BDT 5000–9999 (US$ Σ59–118)	1.8 (1.2 2.6)	0.004
BDT 10,000–14,999 (US$ Σ118–178)	1.2 (0.8 1.9)	0.364
BDT 15,000 and above (US$ Σ178 and above	Ref
CP topography
Monoplegia and hemiplegia	Ref
Diplegia	1.3 (0.8 1.9)	0.269
Triplegia and quadriplegia	0.9 (0.6 1.4)	0.802
GMFCS level
I	Ref
II	0.7 (0.5 1.2)	0.167
III	0.6 (0.3 0.9)	0.027
IV	0.7 (0.4 1.2)	0.192
V	0.4 (0.2 0.7)	0.002
MACS Level
I	Ref
II	0.9 (0.6 1.4)	0.644
III	0.9 (0.6 1.4)	0.570
IV	0.7 (0.4 1.1)	0.148
V	0.8 (0.5 1.4)	0.453
Type of associated impairment^b
Intellectual	1.3 (1.0 1.8)	0.063
Visual	0.9 (0.6 1.4)	0.662
Hearing	2.3 (1.5 3.5)	<0.001

Open in a new tab

^aAll variables found significant in the unadjusted analyses were included in the adjusted model to identify the potential predictors of not receiving rehabilitation services among children with CP in the BCPR.

^bReference category: No impairment.

Discussion

To the best of our knowledge, this is the first population-based study reporting the rehabilitation status and predictors of rehabilitation service uptake among children with CP in an LMIC. We observed that a large number of children with CP in the BCPR had never received any rehabilitation services. Children who had received services were more likely to be female and have educated parents, a higher socio-economic status and severe gross motor impairment.

Similar to this study, poor rehabilitation coverage has been reported among children with CP in India [22]. In contrast, Schmidt et al. [35] reported that 98.9% of children with CP in seven HICs had received rehabilitation services within one year. The proportion of children receiving rehabilitation services varies widely, even between LMICs. A higher proportion of rehabilitation service uptake has been observed in hospital-/institution-based studies, ranging from 55.6% in India [22] to 90.4% in Jordan [13], whereas studies conducted in community-based settings identified considerably poorer rehabilitation service uptake in Uganda (9.7%) [10] and South Africa (26.0%) [25]. The observed differences are most likely due to selection bias in hospital-/institution- and community-based settings and the socioeconomic conditions of study participants (i.e. more affluent people with a higher level of education are more likely to access services).

The age for commencing rehabilitation services among participants was substantially delayed when compared with HICs (3 y 8 mo in Bangladesh vs 1 y 5 mo in Australia) [36]. The reported delays in the diagnosis of CP in rural Bangladesh might play a key role here [7]. Recent evidence suggests that early initiation of rehabilitation services is crucial for the best possible motor outcomes [36].

Among children who had received rehabilitation services in the BCPR cohort, the majority received physiotherapy. Similar to our study, physiotherapy was frequently reported in studies conducted in Jordan (90.4%) [13] and Korea (81.3%) [12]. We found that more than two third of the children registered in the BCPR had MACS level III–V (68.6%) and could have benefitted from occupational therapy. Furthermore, 75.2% of children with speech impairment could have benefitted from speech therapy. However, our findings indicate that none of these children had received the required services. The low number of trained occupational therapists and speech and language therapists compared to physiotherapists (250 vs 260 vs 2400, respectively) with a higher availability in major cities in Bangladesh [37] might be responsible for this disparity. We also found that only 4.7% of children with a GMFCS level III–V had received assistive devices. The poor access to assistive devices found in this study is consistent with earlier studies conducted in LMICs [7,10,24]. Without mobility aids, children with severe functional motor limitations are likely to be bedridden and unable to participate in family, school and community life [10].

We also identified several socio-demographic and clinical factors as barriers in rehabilitation service uptake among our participating children. In terms of gender, female children with CP had a lower likelihood of receiving rehabilitation services when compared to male children. However, the current literature on this issue is conflicting. Young females with CP were found four times more likely to utilise rehabilitation services compared to males in the USA [38]. Whereas Sinha and Sharma [22] reported that there is no relationship between sex and rehabilitation service utilisation of children with CP in India. Women with disabilities face a double burden, because of their gender roles and disabilities, in LMICs [39]. Disability for a female becomes a greater barrier in terms of accessing opportunities such as rehabilitation [39,40]. McConachie et al. [20] described that having a male child can influence parents to seek rehabilitation services, particularly in rural settings. This might be related to the notion that male children need to be able to support a family in the future.

Parental education, and in particular maternal education, was significantly associated with rehabilitation service uptake among children with CP in the BCPR. Children whose parents were literate had significantly higher odds of receiving rehabilitation services. The findings are consistent with studies conducted in India [22] and the USA [38]. It is likely that parents who are educated are more aware of their child’s health condition and needs, and understand the significance of providing rehabilitation to their children with CP. Additionally, parents with less education are more likely to be engaged in daily-basis low-paid jobs, which can make it difficult for them to take time away from work or to cover travel and/or service costs to get their children to rehabilitation centres. Our findings suggest that the majority of children in the BCPR were from impoverished families, and this cohort had higher odds of not receiving rehabilitation services. Financial constraints have been identified as a major barrier to utilising rehabilitation services in studies conducted both in HICs [38] and LMICs [10,22,24]. McConachie et al. [20] describe that a majority of children with CP cannot access rehabilitation services because of the costs, both direct (e.g. rehabilitation service charge) and indirect (e.g. transport cost, accommodation cost, food cost), associated with these services.

In terms of clinical factors, we found that children with GMFCS level III and V had a significantly higher likelihood of receiving rehabilitation services. This result is consistent with earlier studies conducted in Canada [17,26], the USA [26] and Australia [19]. The higher utilisation of rehabilitation services among severely motor-impaired children may be because of their increased rehabilitation needs in order to improve pain, comfort and quality of life. While the importance of rehabilitation for children with GMFCS level III‒V cannot be undervalued, recent evidence suggests that the activity, function and participation of children with GMFCS level I–II could be enhanced through early intervention and rehabilitation services [41].

We also found that children with hearing impairment had a significantly lower probability of receiving rehabilitation services. Similar to our findings, Liljenquist et al. [38] found that children with CP and associated impairments had lower odds of utilising physiotherapy services in the USA. In contrast, Majnemer et al. [17] reported that children with lower intellectual impairment had higher odds of rehabilitation service uptake in Canada. It is not clear why children with hearing impairment have a lower likelihood of receiving rehabilitation in our cohort. Further study is required to investigate the effect of associated impairments on rehabilitation service uptake among children with CP in LMICs like Bangladesh.

Strengths and limitations

This study used population-based data from an established register of children with CP in Bangladesh. Another methodological strength of this research is the adoption of the case definition of CP from the ACPR and the SCPE to ensure international consensus for the clinical diagnosis [6] and measurement of motor functions [42]. Despite our extensive efforts, this study had several limitations, however. Whilst the KIM is cost-effective in the identification of children with disabilities in LMICs [24,43], the BCPR recruitment efforts might have missed some children with CP who have mild motor limitations as the KIM has a 77.6% case-ascertainment rate compared with door-to-door surveying [31]. Therefore, children with severe motor limitations may have been overrepresented in this study. Secondly, the assessment of rehabilitation status was mostly based on the primary caregiver’s responses, due to a lack of service utilisation records. Although there is a chance of recall bias, such a method has previously been used in HICs [17,19] and LMICs [10,24]. Thirdly, due to a lack of medical records, BCPR data collection has to rely on caregiver responses, in addition to the clinical examination, when assessing the severity of associated impairments in some of cases. Although this might have introduced information bias in determining the severity of associated impairments, such methods have also been previously used in other large-scale studies conducted in LMICs and HICs [7,44]. Finally, the national poverty lines were estimated based on household per capita consumption (food and non-food consumption/expenditure) using a detailed questionnaire from the HIES in Bangladesh [34]. Although information with that level of detail is not collected as part of the BCPR, the methodology is similar to some extent. However, there is still a risk of overreporting the poverty level with BCPR data due to the differences in survey tools and depth of information collected compared to the HIES, as well as several other factors.

Conclusions

Nearly half of the children with CP in our study had not have access to rehabilitation services. A majority of children who were in need of assistive devices could not access them. Additionally, the age at commencement of rehabilitation was substantially delayed, limiting the opportunity to improve function and independence. Socio-demographic (i.e. sex, parental education and monthly family income) and clinical factors (i.e. GMFCS level and associated impairments) were significantly associated with rehabilitation service uptake. This evidence has important implications for policy formation and the improvement of rehabilitation services for children with CP in Bangladesh. Locally available and affordable early intervention and rehabilitation service delivery models, including training of rehabilitation professionals regarding community-based management of CP, should be seen as a priority for the strategic development of rehabilitation service coverage among this vulnerable population.

Acknowledgments

We would like to express our heartfelt thanks to all primary caregivers and children with CP for their precious time and voluntary participation in the BCPR. We also would like to cordially thank the CSF Global team in Bangladesh for their diligent work and support in study implementation and guidance to primary caregivers to ensure necessary referral uptake.

Data Availability

The authors are unable to share the de-identified line listed data as the data contain potentially sensitive and identifying patient information; specifically sensitivities around the topic and due to the risk of participant identification given the specific/ defined study location and unique characteristics of participants. This is imposed by the Asian Institute of Disability and Development (AIDD) Human Research Ethics Committee (HREC) as part of the approval for the Bangladesh Medical Research Council ethics. Researchers may contact the AIDD for data access at the following: AIDD, House # 76 & 78, Road # 14, Block B, Banani R/A, Dhaka – 1213, Bangladesh; Phone: +88-02-55040839; Email: disabilityasia@gmail.com.

Funding Statement

This study has been conducted as a part of the BCPR research project. The BCPR is funded by the Research Foundation of Cerebral Palsy Alliance (PG4314 – Bangladesh CP Register) and internal funding from CSF Global, Bangladesh. GK is supported by the Cerebral Palsy Alliance Research Foundation Career Development Fellowship (CDF 0116).

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PLoS One. doi: 10.1371/journal.pone.0250640.r001

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10 Feb 2021

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Rehabilitation status of children with cerebral palsy in Bangladesh: findings from the Bangladesh Cerebral Palsy Register

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Additional Editor Comments:

The paper needs a major revision addressing the comments raised by two independent reviewers.

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Reviewer #1: Yes

Reviewer #2: Yes

**********

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Reviewer #1: Yes

Reviewer #2: Yes

**********

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Reviewer #1: No

Reviewer #2: No

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Reviewer #2: Yes

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Reviewer #1: Title: Rehabilitation status of children with cerebral palsy in Bangladesh: findings from the Bangladesh Cerebral Palsy Register

Objective: To assess the rehabilitation status and factors associated with rehabilitation service utilisation among children with cerebral palsy in Bangladesh.

Overall impression: A well conducted research. Introduction and justification of the study is good. Robust data analysis and rich discussion.

WHAT AUTHORS NEED TO IMPROVE ON

MAJOR

• Line 80 to 104, Line 309, abstract methodology; The paper has stated that data from the Bangladesh CP register was used. However, the methodology involved the researchers identifying the children in the community using key informat method. Data was collected from the children and caregivers and this data was recorded in the CP register.

This is not a study done on analysis of secondary data (Bangladesh CP register), or done from information gathered from secondary data(Bangladesh CP register). Rather, this study used primary data i.e. a community survey to identify children. Data gathered from the survey (from the children and care givers) was imputed into the BCPR.

Saying the study used data from Bangladesh CP Register (BCPR) is misleading (this portrays a study done by analyzing secondary data). This study correctly put is a POPULATION BASED STUDY DONE AMONG CP CHILDREN IN BANGLADESH. In addition to carrying out the study, data generated from the survey was imputed into the BCPR. This should be corrected in all aspects of the manuscript, including the abstract.

• Table 3 and Table 4. Please redo the bivariate analysis(chi square) using row total instead of column total. That way, the results will tally with the unadjusted odds ratio reported alongside.

• Table 3 and Table 4. Please indicate clearly on the COLUMN OF ‘UNADJUSTED OR’ if the outcome is ‘RECEIVED REHABILITATION’ or ‘NOT RECEIVED REHABILITATION’. This will make interpretation of the tables better and simpler,

Reviewer #2: Thank you for the paper. This is an important area of work in cerebral palsy research. However, there is need for some clarity on methods and results. See specific comments below.

Some additional point:

Introduction

• Very well written

Methods

• Provide some details of the Bangladesh CP Register (BCPR). How long has it been established for? How was surveillance area identified? Who collects data? Who is responsible for data management? Is data available publicly?

• Provide details of key informant method to enroll children into the registry.

• Page 6, line 104: Medical records reviewed were with primary caregivers or were they reviewed at the facilities where children received rehabilitation?

• Page 6, line 107: What data source was used for data on general population?

• What was the main outcome variable? Add details on how it is collected in BCPR.

• Provide details on how variables added to the analysis were handled.

• How was poverty level assessed?

Results

• Clarify “Illiterate mothers had 2.1 times (95% CI 1.8-2.5) less chance of receiving rehabilitation services” whether this relates to mothers receiving rehabilitation services of children of mothers with no education.

• Clarify “Children with a monthly income of BDT 5000-9999 (US$ ⁓59-118) were 1.7 [95% CI 1.5-2.0] times less likely to receive rehabilitation services than children with a monthly family income of BDT 15000 (US$ ⁓178) and above”. This is family or household income, not child’s income. Recheck.

Discussion

• Nicely written

General

• Mention full form at the first mention of an abbreviation

• Check format of the paper so that it is in accordance with the journal’s guidelines

• Check references, their formatting and citation style as per the journal’s requirements

• Check for language edits and sentence structure

Thank you.

**********

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Reviewer #1: No

Reviewer #2: Yes: Nukhba Zia

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PLoS One. 2021 May 3;16(5):e0250640. doi: 10.1371/journal.pone.0250640.r002

Author response to Decision Letter 0

13 Mar 2021

Professor Joerg Heber

Editor-in-Chief

PLOS ONE

RE: Manuscript ID PONE-D-21-00848

Title: Rehabilitation status of children with cerebral palsy in Bangladesh: findings from the Bangladesh Cerebral Palsy Register

We would like to thank the academic editor and the reviewers for their constructive feedback and helpful comments on our manuscript titled ‘Rehabilitation status of children with cerebral palsy in Bangladesh: findings from the Bangladesh Cerebral Palsy Register’. Please see below our point-to-point responses to the academic editor’s and reviewers’ comments.

Academic Editor:

Comment-1: Please ensure that your manuscript meets PLOS ONE's style requirements, including those for file naming. The PLOS ONE style templates can be found at

https://journals.plos.org/plosone/s/file?id=wjVg/PLOSOne_formatting_sample_main_body.pdf and

https://journals.plos.org/plosone/s/file?id=ba62/PLOSOne_formatting_sample_title_authors_affiliations.pdf

Our response: Thank you for the feedback. We have revised the title page and main body of the manuscript to meet the PLOS ONE style requirements.

Comment-2: Thank you for describing the provision of informed participant consent in the Methods section of your manuscript. We ask that you additionally provide this information in the Ethics Statement.

Our response: Thank you for the appreciation and recommendation. We have now added the following statement in the Ethics Statement of the submission system.

‘Written informed consent was given by parents or primary caregivers of all study participants prior registration into the BCPR and data collection.’

Comment-3: We note that you have indicated that data from this study are available upon request. PLOS only allows data to be available upon request if there are legal or ethical restrictions on sharing data publicly. For information on unacceptable data access restrictions, please see http://journals.plos.org/plosone/s/data-availability#loc-unacceptable-data-access-restrictions.

In your revised cover letter, please address the following prompts:

We will update your Data Availability statement on your behalf to reflect the information you provide.

Our response: Thank you for the suggestions. The authors are unable to share the de-identified line listed data as the data contain potentially sensitive and identifying patient information; specifically sensitivities around the topic and due to the risk of participant identification given the specific/ defined study location and unique characteristics of participants. This is imposed by the Asian Institute of Disability and Development (AIDD) Human Research Ethics Committee (HREC) as part of the approval for the Bangladesh Medical Research Council ethics. Researchers may contact the AIDD for data access at the following: AIDD, House # 76 & 78, Road # 14, Block B, Banani R/A, Dhaka – 1213, Bangladesh; Phone: +88-02-55040839; Email: disabilityasia@gmail.com.

Comment-4: PLOS requires an ORCID iD for the corresponding author in Editorial Manager on papers submitted after December 6th, 2016. Please ensure that you have an ORCID iD and that it is validated in Editorial Manager. To do this, go to ‘Update my Information’ (in the upper left-hand corner of the main menu), and click on the Fetch/Validate link next to the ORCID field. This will take you to the ORCID site and allow you to create a new iD or authenticate a pre-existing iD in Editorial Manager. Please see the following video for instructions on linking an ORCID iD to your Editorial Manager account: https://www.youtube.com/watch?v=_xcclfuvtxQ

Our response: Thank you for the suggestion. The ORCID iD of the corresponding author has been added.

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We require you to either (1) present written permission from the copyright holder to publish these figures specifically under the CC BY 4.0 license, or (2) remove the figures from your submission:

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The following resources for replacing copyrighted map figures may be helpful:

USGS National Map Viewer (public domain): http://viewer.nationalmap.gov/viewer/

The Gateway to Astronaut Photography of Earth (public domain): http://eol.jsc.nasa.gov/sseop/clickmap/

Maps at the CIA (public domain): https://www.cia.gov/library/publications/the-world-factbook/index.html and https://www.cia.gov/library/publications/cia-maps-publications/index.html

NASA Earth Observatory (public domain): http://earthobservatory.nasa.gov/

Landsat: http://landsat.visibleearth.nasa.gov/

USGS EROS (Earth Resources Observatory and Science (EROS) Center) (public domain): http://eros.usgs.gov/#

Natural Earth (public domain): http://www.naturalearthdata.com/

Our response: Thank you for the detailed information and kind suggestion on this important issue. Fig 1 of the manuscript illustrates the study sites of the BCPR along with the number of children recruited into the BCPR from different districts of Bangladesh. We (authors) produced this map using ArcGIS Desktop 10.8 software. We assure you that there is no copyright issue associated with the map. Thus, this map can be made freely available online, and any third party is permitted to access, download, copy, distribute, and use the map in any way, even commercially, with proper attribution. This information has now been added into the methodology section of the revised manuscript. (Please see line 93 and 94 of the unmarked manuscript)

Comment-6: The paper needs a major revision addressing the comments raised by two independent reviewers.

Our response: Thank you for the feedback. We have revised the manuscript incorporating all comments of the reviewers.

Reviewer: 1

Comment-1:

Title: Rehabilitation status of children with cerebral palsy in Bangladesh: findings from the Bangladesh Cerebral Palsy Register

Objective: To assess the rehabilitation status and factors associated with rehabilitation service utilisation among children with cerebral palsy in Bangladesh.

Overall impression: A well conducted research. Introduction and justification of the study is good. Robust data analysis and rich discussion.

Our response: Thank you for your kind comments.

Comment-2: Line 80 to 104, Line 309, abstract methodology; The paper has stated that data from the Bangladesh CP register was used. However, the methodology involved the researchers identifying the children in the community using key informat method. Data was collected from the children and caregivers and this data was recorded in the CP register.

Our response: Thank you for the valuable suggestion. We have incorporated the suggestion and edited the manuscript accordingly. (Please see line 5-7, 79-82 of the unmarked manuscript)

Comment-3: Table 3 and Table 4. Please redo the bivariate analysis(chi square) using row total instead of column total. That way, the results will tally with the unadjusted odds ratio reported alongside.

Our response: Thank you for the feedback. We have revised the Table 3 and Table 4 and added row percentages instead of column percentages. (Please see the Table 3 and Table 4 on page 12-14)

Comment-4: Table 3 and Table 4. Please indicate clearly on the COLUMN OF ‘UNADJUSTED OR’ if the outcome is ‘RECEIVED REHABILITATION’ or ‘NOT RECEIVED REHABILITATION’. This will make interpretation of the tables better and simpler.

Our response: Thank you for the suggestion. As recommended, we have inserted ‘Unadjusted odds ratios for not receiving rehabilitation’ in Table 3 and Table 4. (Please see the Table 3 and Table 4 on page 12-14)

Reviewer-2:

Comment-1: Thank you for the paper. This is an important area of work in cerebral palsy research. However, there is need for some clarity on methods and results.

See specific comments below.

Our response: Thank you for your constructive feedback to improve our paper.

Comment-2: Introduction: Very well written

Our response: Thank you for the appreciation.

Comment-3: Methods: Provide some details of the Bangladesh CP Register (BCPR). How long has it been established for?

Our response: Thank you for the valuable comment. We established the first population-based surveillance of children with CP i.e. Bangladesh CP Register (BCPR) in rural Bangladesh in 2015. The BCPR is an ongoing surveillance that studies epidemiology, rehabilitation, intervention strategies to improve functional outcomes and limit associated impairments among children with CP in Bangladesh [Khandaker et al. 2015]. Initially, the surveillance activities (i.e. BCPR) were confined in one subdistrict (Shahjadpur, ~325 square kilometres, ~123,576 households) which represents rural and semi-urban Bangladesh (where the majority of the population (76.7%) lives [Bangladesh Bureau of Statistics, 2014]) in terms of demographic and other indicators (e.g. birth rate, immunisation rate, perinatal mortality rate, literacy rate). To date, this population-based surveillance has been maintained with high case ascertainment in Shahjadpur. Additionally, between 2015 and 2019 the BCPR was scaled up to 17 other subdistricts (⁓4,338 square kilometres, ~1,304,960 households) following opportunistic recruitment at the community level (Fig 1). (Please see line 79-90 of the unmarked manuscript)

References:

Khandaker G, Smithers-Sheedy H, Islam J, Alam M, Jung J, Novak I, et al. Bangladesh Cerebral Palsy Register (BCPR): a pilot study to develop a national cerebral palsy (CP) register with surveillance of children for CP. BMC Neurol. 2015;15:173. Epub 2015/09/27. doi: 10.1186/s12883-015-0427-9. PubMed PMID: 26407723; PubMed Central PMCID: PMCPMC4582618.

Bangladesh Bureau of Statistics. Bangladesh Population and Housing Census 2011. Dhaka, Bangladesh: Bangladesh Bureau of Statistics. 2014; [cited 2020 Aug]. Available from: http://203.112.218.65:8008/WebTestApplication/userfiles/Image/National%20Reports/Union%20Statistics.pdf

Comment-4: How was surveillance area identified?

Our response: Thank you for the query. Our population-based surveillance area (i.e. Shahjadpur) was selected after assessing local demographic and other indicators (e.g. birth rate of Shahjadpur vs. Bangladesh, immunization of Shahjadpur vs. Bangladesh, perinatal mortality rate of Shahjadpur vs. Bangladesh, literacy of Shahjadpur vs. Bangladesh,). We wanted to select a sub-district that is most representative of rural and semi-urban areas of Bangladesh as 76.7% of areas are rural [Bangladesh Bureau of Statistics, 2014]. Moreover, we had existing community engagement and ongoing projects in the same community since 2003 which helped us to maintain a high case ascertainment rate in the surveillance area. All of these factors were considered while choosing the surveillance site for the BCPR. This information has now been added to the methodology section of the revised manuscript. (Please see line 82-88 of the unmarked manuscript)

Reference:

Comment-5: Who collects data? Who is responsible for data management?

Our response: Thank you for the valuable comment. As part of registration into the BCPR, data on selected variables were collected using a standard case record form adopted from the Australian CP Register by interviewing the primary caregivers, clinical assessment, reviewing medical records (if available). In the current analyses, we used the following variables: (i) socio-demographic characteristics (e.g. age, gender, educational level of parents, monthly family income); (ii) clinical characteristics (e.g. Gross Motor Function Classification System (GMFCS) level, Manual Ability Classification System (MACS) level, predominant CP motor type, CP topography and associated impairments) and (iii) information on rehabilitation (i.e. whether the child ever received rehabilitation, the type and location/source of services received, age of commencement of rehabilitation).

Following data collection data were entered electronically into the password-protected BCPR online data repository (i.e. http://bangladesh.cpregister.com/) with access to named investigators only. A dedicated data management team located at CSF Global in Dhaka, Bangladesh with support from investigators conducts the data management and analyses. This information has now been added to the methodology section of the revised manuscript. (Please see line 111-128 of the unmarked manuscript)

Comment-6: Is data available publicly?

Our response: Thank you for the query. The BCPR data is not available publicly. The BCPR data contain potentially sensitive and identifying information of children with CP and their primary caregivers. This information has been added to the Data Availability Statement.

Comment-7: Provide details of key informant method to enroll children into the registry.

Our response: Thank you for the valuable feedback. As part of the ongoing surveillance (i.e. BCPR), children with suspected CP in a community are identified using the key informant method (KIM). The KIM is a validated method where local volunteers (e.g. religious leaders, teachers, community health workers etc.) are trained as key informants (KIs) to identify children with disabilities in their communities [Mackey et al. 2012]. The KIs are identified (approximately 1 KI per village) by the Community Mobilisers (CMs—paid project staff) and receive a day-long training on the identification of children with suspected CP, disability sensitisation, advocacy for a disability inclusive society using flip charts, group work and role play. Following the training, the KIs are provided 4–6 weeks to identify and enlist children with suspected CP and share their contact details with CMs to bring those children and their primary caregivers to the nearest medical assessment camps for a confirmed diagnosis, detailed neurodevelopmental assessments and registration in the BCPR. The clinical definition adopted from the Surveillance of CP in Europe (SCPE) [Surveillance of cerebral palsy in Europe, 2000] and the Australian CP Register (ACPR) [Australian Cerebral Palsy Register Group, 2018] are strictly followed during case ascertainment. As suggested, we have now added the details with relevant references in our revised manuscript. (Please see line 96-110 of the unmarked manuscript)

References:

Australian Cerebral Palsy Register Group. Report of the Australian Cerebral Palsy Register, birth years 1995-2012; 2018. Australia; [cited 2020 Aug]. Available from: https://www.ausacpdm.org.au/resources/ australian-cerebral-palsy-register/

Mackey S, Murthy GV, Muhit MA, Islam JJ, Foster A. Validation of the key informant method to identify children with disabilities: methods and results from a pilot study in Bangladesh. J Trop Pediatr. 2012;58(4):269-74. Epub 2011/11/15. doi: 10.1093/tropej/fmr094. PubMed PMID: 22080830.

Surveillance of cerebral palsy in Europe. Surveillance of cerebral palsy in Europe: a collaboration of cerebral palsy surveys and registers. Surveillance of Cerebral Palsy in Europe (SCPE). Dev Med Child Neurol. 2000;42(12):816-24. Epub 2000/12/29. doi: 10.1017/s0012162200001511. PubMed PMID: 11132255.

Comment-8: Page 6, line 104: Medical records reviewed were with primary caregivers or were they reviewed at the facilities where children received rehabilitation?

Our response: Thank you for the query. The medical records available with primary caregivers were reviewed during medical assessment camps. The statement has been edited accordingly. (Please see line 126-128 of the unmarked manuscript)

Comment-9: Page 6, line 107: What data source was used for data on general population?

Our response: Thank you for the query. The data on age, sex and education of the general population were collected from the Bangladesh Demographic and Health Survey (BDHS), 2014 [National Institute of Population Research and Training (NIPORT), Mitra and Associates and ICF International, 2016]. The data on families below the international poverty line among the general population were collected from the Household Income and Expenditure Survey 2016 [Bangladesh Bureau of Statistics, 2019]. These data sources are mentioned in the Table 1 footnotes as well as in the methodology section of the revised manuscript. (Please see line 145-147, 186, 187 and 189 of the unmarked manuscript)

References:

National Institute of Population Research and Training (NIPORT), Mitra and Associates, ICF International. Bangladesh Demographic and Health Survey 2014. Dhaka, Bangladesh, and Rockville, Maryland, USA: NIPORT, Mitra and Associates, and ICF International: 2016. [cited 2020 Aug]. Available from: https://dhsprogram.com/pubs/pdf/FR311/FR311.pdf

Bangladesh Bureau of Statistics. Preliminary Report on Household Income and Expenditure Survey 2016. Dhaka, Bangladesh: Bangladesh Bureau of Statistics. 2019; [cited 2020 Aug]. Available from: http://bbs.portal.gov.bd/sites/default/files/files/bbs.portal.gov.bd/page/b343a8b4_956b_45ca_872f_4cf9b2f1a6e0/HIES%20Preliminary%20Report%202016.pdf

Comment-10: What was the main outcome variable? Add details on how it is collected in BCPR.

Our response: Thank you for the query. The main outcome variable was the rehabilitation status (i.e. whether he/she ever received rehabilitation) of a child with CP registered in the BCPR, which is a binary variable with ‘yes’ and ‘no’ as responses. Information on rehabilitation was collected from primary caregivers by a physiotherapist during medical assessment camps in the context of BCPR recruitment. To document the type of rehabilitation services received, multiple responses were allowed and, therefore, the numbers presented for this variable are not mutually exclusive. Additionally, the medical records available from primary caregivers were reviewed for any documentation of rehabilitation services. The information has been elaborated in the methodology. (Please see line 120-128 of the unmarked manuscript)

Comment-11: Provide details on how variables added to the analysis were handled.

Our response: Thank you for the query. To maintain the quality of the data, we used a double data entry method. Subsequently, data-entry error checks were performed by running frequencies of all variables to identify any outliers. In case of any missing data or incorrect/suspicious information, the BCPR case record forms (i.e. source documents) were reviewed. If the information was not found in the BCPR form, it was relayed to the field team and where possible the participants were contacted to gather the missing information. Continuous variables were collected as exact values and later recoded and categorised into groups (e.g. age was categorised as follows: 0–4 years, 5–9 years, 10–14 years and 15–18 years). Similarly, the monthly family income data were converted to United States Dollar (US$) (considering 1 US$ = 84.43 Bangladeshi taka (BDT)) and categorised into four family-income groups (i.e. BDT 500–4999 (US$ ⁓6–59), BDT 5000–9999 (US$ ⁓59–118), BDT 10,000–14,999 (US$ ⁓118–178) and BDT 15,000 and above (US$ ⁓178 and above). Comparison between the BCPR cohort and the general population was performed using the 2014 Bangladesh Demographic and Health Survey (BDHS) [National Institute of Population Research and Training (NIPORT), Mitra and Associates and ICF International, 2016] and Household Income and Expenditure Survey [Bangladesh Bureau of Statistics, 2019] data from 2016. The poverty level among the families in the BCPR cohort was estimated using the national poverty lines (at the divisional level) as a cut-off. The proportion of families living below the poverty lines was then compared with the general population of the respective divisions as reported in the Household Income Expenditure Survey (HIES) in Bangladesh [Bangladesh Bureau of Statistics, 2019]. Bivariate analyses were completed to assess the impact of socio-demographic and clinical factors on rehabilitation status. For regression models, ‘Not receiving rehabilitation’ was considered as the main outcome of interest. Factors that were found to be statistically significant in unadjusted logistic regression were fitted into an adjusted logistic regression model. Odds ratios with 95% confidence intervals (CI) were reported. A p value <0.05 was considered significant. All data were analysed using the Statistical Package for the Social Sciences (SPSS) software, version 26 (IBM, Armonk, NY, USA).

The information on data management and analysis has been elaborated in the methodology section. (Please see line 134-158 of the unmarked manuscript)

References:

Comment-12: How was poverty level assessed?

Our response: Thank you for the query. In the BCPR surveillance we collect information related to the household income-expenditure in local currency (i.e. Bangladesh Taka (BDT)) by probing the major food and non-food consumption categories. To estimate the proportion of families living below the poverty line we previously used the international poverty line (US$ 1.90 per day per capita) as a cutoff value. Since we have submitted this manuscript, we have carefully looked into the poverty data. Subsequently, we have identified some additional concern which might worth considering in terms of poverty/income data for our cohort. For instance, the international poverty line US$ 1.90 per day per capita is an absolute poverty line in purchasing power parity (PPP) to allow comparison with other countries [The World Bank, 2019] which is estimated based on the national poverty lines of selected countries. This conversion of the poverty lines is complex and requires consideration of multiple factors as well as careful interpretation [Jolliffe and Prydz, 2016]. Considering the complexity and risk of misinterpretation, we have now excluded this variable (i.e. International poverty line) from our analysis. Instead, we have used the national poverty line (at the divisional level) of Bangladesh to report the proportion of families living below the poverty line. Please see our revised analysis in the Table 1 and the result section (Please see line 178-182 of the unmarked manuscript). We have also added this information in the methodology section of the revised manuscript. (Please see line 147-151 of the unmarked manuscript)

The national poverty lines are estimated based on household per capita consumption (food and non-food consumption/expenditure) using a detailed questionnaire during the Household Income Expenditure Survey (HIES) in Bangladesh. Though we did not collect that detailed information as part of the BCPR, our methodology is to some extent similar. However, there is a risk of overreporting the poverty level in the BCPR due to the differences in survey tools and depth of information collected compared to the HIES, and several other factors. We have now added this information in the limitation section of our manuscript. (Please see line 370-376 of the unmarked manuscript)

References:

The World Bank. How is the global poverty line derived? How is it different from national poverty lines? The World Bank; 2020. Available from: https://datahelpdesk.worldbank.org/knowledgebase/articles/193310-how-is-the-global-poverty-line-derived-how-is-it

Jolliffe D, Prydz EB. Estimating international poverty lines from comparable national thresholds. The World Bank; 2016 Mar 17.

Comment-13:

Results:

Our response: Thank you for the constructive feedback. The sentences have been revised as follows:

‘Children of mothers with no education had 2.1 times (95% CI 1.8-2.5) less chance of receiving rehabilitation services’. (Please see line 224-226 of the unmarked manuscript) ‘Children from families with a monthly income of BDT 5000-9999 (US$ ⁓59-118) were 1.7 [95% CI 1.5-2.0] times less likely to receive rehabilitation services than children with a monthly family income of BDT 15000 (US$ ⁓178) and above’. (Please see line 232-234 of the unmarked manuscript)

Comment-14: Discussion: Nicely written.

Our response: Thank you for the appreciation.

Comment-15: Mention full form at the first mention of an abbreviation

Our response: Thank you for the feedback. We have reviewed the manuscript thoroughly and confirmed to use the abbreviations only after stating the full form at the first mention.

Comment-16: Check format of the paper so that it is in accordance with the journal’s guidelines

Our response: Thank you for the feedback. There were some formatting errors that have been corrected in the revised manuscript.

Comment-17: Check references, their formatting and citation style as per the journal’s requirements

Our response: Thank you for the feedback. The in-text citations and bibliography have been revised following the journal guidelines.

Comment-18: Check for language edits and sentence structure

Our response: Thank you for the feedback to improve the readability of our manuscript. The manuscript has been reviewed and edited by a professional proofreader.

Yours sincerely,

Gulam Khandaker

On behalf of the study investigators

Attachment

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PLoS One. doi: 10.1371/journal.pone.0250640.r003

Decision Letter 1

Enamul Kabir

31 Mar 2021

PONE-D-21-00848R1

Rehabilitation status of children with cerebral palsy in Bangladesh: findings from the Bangladesh Cerebral Palsy Register

PLOS ONE

Dear Dr. Khandaker,

Please address comments from one of the reviewers.

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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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6. Review Comments to the Author

Reviewer #1: In the methods section, line 96 to line 133 should be written in past tense. Read through the methods section to ensure its written in the correct tenses.

Reviewer #2: Thank you for making the revisions and addressing all comments raised based on the last version of the manuscript. Thanks

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Reviewer #1: Yes: Dr Tope Olubodun

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PLoS One. 2021 May 3;16(5):e0250640. doi: 10.1371/journal.pone.0250640.r004

Author response to Decision Letter 1

5 Apr 2021

05 April 2021

Professor Joerg Heber

Editor-in-Chief

PLOS ONE

RE: Manuscript ID PONE-D-21-00848

Title: Rehabilitation status of children with cerebral palsy in Bangladesh: findings from the Bangladesh Cerebral Palsy Register

We would like to thank the academic editor and the reviewers for their constructive feedback on our manuscript titled ‘Rehabilitation status of children with cerebral palsy in Bangladesh: findings from the Bangladesh Cerebral Palsy Register’. We have incorporated all of the Academic Editor’s and Reviewers’ feedback into our revised manuscript. Additionally, all statistical methods employed in this study were re-performed and reviewed. The errors and inconsistencies identified during the review have been corrected. Please see below our point-to-point responses to the editor’s and reviewers’ comments and a detailed description of the additional changes that we have made as part of the rebuttal.

Editor:

Comment-1: 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.

Our response: Thank you for the feedback. We have reviewed the reference list. There were some errors in the reference list (e.g. place of publication in reference# 1, 30, 33 and 34; semicolon in reference# 1, 6, 30, 33, 34; erratum in reference#3, 5 and 36; DOI in reference# 4, 8, 9, 20, 24, 27, 38, 41, 42 and 44; publisher name in reference# 40; book chapter format in reference# 11 and 39; author list in reference# 27 and journal name abbreviation in reference# 43) that have been corrected in the revised version of the manuscript. Sincere apologies for the unintentional errors. We confirm that we have not cited any retracted article in this manuscript.

Reviewer: 1

Comment-1:

In the methods section, line 96 to line 133 should be written in past tense. Read through the methods section to ensure its written in the correct tenses.

Our response: Thank you for the valuable comment. We have revised the methodology section to incorporate the suggestion. Please see lines 96-133.

Additional changes:

As part of this rebuttal, to ensure statistical integrity of the study findings, all statistical procedures were re-performed and reviewed. We subsequently identified that the method of computing dummy variable for age categories (i.e. Age groups 5-9 and 10-14) was incorrect and there was a calculation error in the ‘Number of associated impairments’ variable. As we have corrected these two variables, the unadjusted odds ratios for these variables (Table 3 [row 4 and 5] and Table 4 [row 30-32]) and adjusted odds ratios and corresponding p values for all variables entered into the multiple regression model (Table 5) have also changed to some extent. Considering the corrections, we have now revised the abstract (line 16-23), results (line 260, 267 and 271-277), discussions (line 289, 322-332 and 348-353) and conclusions (line 392) sections of the manuscript (unmarked version) accordingly. Additionally, the lower limit of 95% Confidence Interval of unadjusted odds ratios for Hearing Impairment has been changed from 1.7 to 1.6 (Table 4 [row 27]). We sincerely apologise for these unintentional errors that we have corrected now. However, we would like to confirm that the revised results have not made any major changes to our study findings.

Yours sincerely,

Gulam Khandaker

On behalf of the study investigators

Attachment

Submitted filename: Rebuttal Letter_BCPR Rehab_05.04.21_V2.docx

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PLoS One. doi: 10.1371/journal.pone.0250640.r005

Decision Letter 2

Enamul Kabir

12 Apr 2021

Rehabilitation status of children with cerebral palsy in Bangladesh: findings from the Bangladesh Cerebral Palsy Register

PONE-D-21-00848R2

Dear Dr. Khandaker,

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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Kind regards,

Enamul Kabir

Academic Editor

PLOS ONE

Additional Editor Comments (optional):

Reviewers' comments:

PLoS One. doi: 10.1371/journal.pone.0250640.r006

Acceptance letter

Enamul Kabir

23 Apr 2021

PONE-D-21-00848R2

Rehabilitation status of children with cerebral palsy in Bangladesh: findings from the Bangladesh Cerebral Palsy Register

Dear Dr. Khandaker:

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

Dr. Enamul Kabir

Academic Editor

PLOS ONE

Associated Data

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

Supplementary Materials

Attachment

Submitted filename: Rebuttal letter_BCPR Rehab_V1.doc

Click here for additional data file.^{(213KB, doc)}

Attachment

Submitted filename: Rebuttal Letter_BCPR Rehab_05.04.21_V2.docx

Click here for additional data file.^{(30.2KB, docx)}

Data Availability Statement

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PERMALINK

Rehabilitation status of children with cerebral palsy in Bangladesh: Findings from the Bangladesh Cerebral Palsy Register

Mahmudul Hassan Al Imam

Israt Jahan

Manik Chandra Das

Mohammad Muhit

Hayley Smithers-Sheedy

Sarah McIntyre

Nadia Badawi

Gulam Khandaker

Roles

Abstract

Objective

Materials and methods

Results

Conclusions

Introduction

Materials and methods

Fig 1. Map of Bangladesh demonstrating the distribution of BCPR-registered children from each district in the study sample.

Study participants and data collection method

Data management and analysis

Ethical considerations

Results

Socio-demographic characteristics

Table 1. Socio-demographic characteristics of children with CP.

Rehabilitation status

Table 2. Rehabilitation status of children with CP in the BCPR cohort.

Factors influencing receipt of rehabilitation services among children with CP

Age and sex of children with CP

Table 3. Socio-demographic factors related to the receipt of rehabilitation for children with CP in the BCPR cohort.

Education of parents

Monthly family income

Predominant motor type and topography of CP

Table 4. Clinical factors related to the receipt of rehabilitation for children with CP in the BCPR cohort.

GMFCS and MACS level

Associated impairments

Independent predictors of not receiving rehabilitation services among children with CP in the BCPR cohort

Table 5. Predictors of not receiving rehabilitation service for children with CP in the BCPR cohort.

Discussion

Strengths and limitations

Conclusions

Acknowledgments

Data Availability

Funding Statement

References

Decision Letter 0

Enamul Kabir

Roles

Author response to Decision Letter 0

Decision Letter 1

Enamul Kabir

Roles

Author response to Decision Letter 1

Decision Letter 2

Enamul Kabir

Roles

Acceptance letter

Enamul Kabir

Roles

Associated Data

Supplementary Materials

Data Availability Statement

ACTIONS

PERMALINK

RESOURCES

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