OSA 18 Questionnaire: Tool to Evaluate Quality of Life and Efficacy of Treatment Modalities in Pediatric Sleep Disordered Breathing Due to Adenotonsillar Hypertrophy

Abstract

Aims to evaluate quality of life in paediatric SDB due to adenotonsillar hypertrophy and efficacy of treatment modalities (medical and surgical) by using OSA-18 questionnaire. Prospective study, conducted from April 2019 to June 2019, including 42 patients with clinical features suggestive of SDB due to adenotonsillar hypertrophy, in age group of 3–15 years. Nasopharyngoscopy was done to grade adenoid hypertrophy. OSA-18 QOL questionnaire was recorded in all patients and depending upon the severity of impact of QOL and grades of adenoid hypertrophy, patients were categorized into two groups. Group 1 received medical treatment and group 2 underwent adenotonsillectomy. Questionnaire was again recorded after 4 weeks. Pretreatment and post-treatment total mean and individual domain scores were compared. Paired t tests was used to evaluate results. Group 1 included 16 children with mild to moderate impact and received medical management. Pretreatment mean OSA-18 score of 70.31 was improved to 33.5. Group 2 enrolled 26 patients with severe impact, were subjected to adenotonsillectomy. Pretreatment and post-treatment mean score were 95.88 and 24.92 respectively. Both groups showed statistically significant improvement in all individual domains and total mean OSA-18 scores indicating improvement in QOL after treatment and efficacy of medical management for mild-moderate SDB and surgery for severe cases. OSA-18 questionnaire is self-administered and disease specific screening tool for early diagnosis and evaluation of QOL before and after treatment. It also helps to categorize patients for advocating appropriate treatment and to evaluate efficacy of treatment modalities.

Introduction

Sleep-disordered breathing (SDB), comprises 10–50% of practice for paediatric otolaryngologists [1], is caused by a complex pathophysiological process and refers to a broad spectrum comprising of primary snoring, upper airway resistance syndrome (UARS) obstructive hypoventilation syndrome and obstructive sleep apnea (OSA). The prevalence of snoring ranges from 1 to 15% out of which habitual snoring averages 5% [2–4]. Prevalence of OSA, as documented by polysomnography (PSG), is 1–4% with peak between age of 2–8 years [2, 5]. OSA, most severe amongst SDB, is characterized by repeated hypoxia and hypercapnia which lead to production of proinflammatory cytokines and induction oxidative stress leading to cellular injury and death [6]. OSA is associated with significant morbidities like; impairment of neurocognitive development and school performance, behavioral problems (including hyperactivity), cardiovascular morbidities, metabolic consequences. SDB increases children’s all cause morbidity by a factor of 6.58 and nearly doubles their health-care utilization as compared with their peers, with more hospital visits and more medications for respiratory infections [7, 8]. Long standing pediatric SDB leads to significant clinical impact and may affect patient quality of life (QOL). Although complications of untreated SDB can harm a child’s health in the long term, they are by enlarge are reversible with treatment.

Adenotonsillar hypertrophy is the most common cause of paediatric SDB. Literature has witnessed the positive impact of Adenotonsillectomy (AT) on the QOL, PSG variables and behavioral sequelae [9–13]. Recently, anti-inflammatory medications such as nasal corticosteroids spray, systemic antihistamines and the leukotriene modifier (montelukast) are used for treatment of mild SDB due to adenotonsillar hypertrophy and showed improvement in terms of apnea–hypopnea index (AHI) and nocturnal oxygen saturation [14–19].

The gold standard test for diagnosis of OSA and assessment of its severity is an overnight, attended, in laboratory polysomnography (PSG) with end tidal CO₂. Pediatric PSG is not widely and easily available, costly and time consuming. It also fails to define the cause of OSA and to quantify the impact of OSA on a child’s general well-being and QOL, including emotional and behavioral health. Most of the otolaryngologists, in their routine practice, diagnose SDB with history, clinical features and nasal endoscopy or x-ray nasopharynx. PSG is reserved in special cases of obesity, craniofacial anomalies, syndromic child, sickle cell disease and mucopolysaccharidosis. But accuracy of history and clinical examination for diagnosing SDB is very low. An alternative evaluation tool should be developed for bridging the gap between clinical history—examination and PSG for diagnosis of SDB. Questionnaires which evaluate the impact on QOL and its improvement after treatment and include symptoms and signs of SDB were developed to aid early diagnosis.

Obstructive sleep apnea 18-items quality of life questionnaire (OSA-18) is a most widely used disease specific QOL survey and a potentially useful screening tool for diagnosis of pediatric OSA. It includes questions directed towards patient’s signs, symptoms, consequences and parent’s concern [20–23]. Franco et al. [20] and Kang et al. [24] had confirmed significant correlation between the OSA-18 scores and PSG parameters and hence validated OSA-18 questionnaire as a diagnostic screening tool. In addition to clinical diagnosis, it may help to identify patients who are appropriate candidates for treatment or failure of treatment or associated symptoms that fail to improve after treatment. Numerous studies [21–23] had demonstrated efficacy of OSA-18 questionnaire in evaluating QOL and its improvement after Adenotonsillectomy which in turn assess its efficacy for SDB. Literature has demonstrated role of medical management for mild SDB, considering surgical risk and caregiver’s anxiety regarding surgery [16–19].

Tonsils and adenoids of grades III and IV can be a primary cause of OSA and their sizes are positively related to risk and severity of OSA [25, 26]. Therefore, tonsil and adenoid size should be considered when selecting treatment options.

In our study, we are using OSA-18 questionnaire scores and adenoid hypertrophy as a measure for severity of SDB. Depending upon the severity of SDB (OSA-18 scores) and sizes of adenoid, treatments were individualized as surgical or medical. Efficacy of the treatment modality was also assessed by using OSA-18 questionnaire.

Aims and Objectives

1. To individualize treatment for SDB by using OSA-18 scores and grades of adenoid hypertrophy.

2. To evaluate impact of SDB due to adenotonsillar hypertrophy on QOL by using OSA-18 questionnaire.

3. To evaluate efficacy of treatment modalities (medical and surgical) by using OSA-18 questionnaire.

Materials and Methods

The prospective (interventional) study conducted in Department of ENT, Star Hospital, Hyderabad with prior Ethical committee approval. Study included 42 patients in age range of 3–18 years, presented with clinical features suggestive of SDB (Table 1 [27]). Study was conducted from April 2019 to June 2019.

Table 1.

Symptoms and signs of sleep disordered breathing

Frequent snoring (≥ 3 nights/week)

Mouth breathing

Labored breathing during sleep

Gasps/snorting noises/observed episodes of apnea

Cyanosis

Sleep enuresis (especially secondary enuresis)

Sleeping in a seated position or with the neck hyperextended

Restless sleep

Headaches on awakening

Daytime sleepiness

Attention-deficit/hyperactivity disorder

Learning problems/poor concentration

Underweight or overweight

Adenoid and tonsilar hypertrophy

Adenoid facies

Inclusion criteria—children with clinical features of SDB [27] and evidence of adenotonsillar hypertrophy.

Exclusion Criteria—craniofacial anomalies, syndromic children, neuromuscular disorders, obesity, mental retardation, history of adenotonsillectomy, parents not willing to participate in study.

All patients were subjected to detailed history (dictated by parents), local and systemic examination. Weight, height and BMI were noted. Adenoid hypertrophy was classified as per flexible fiber optic endoscopic findings; Grade 1-adenoids occupying less than 25% of choanal area; Grade 2-occupying 25–50%; Grade 3-occupying 50–75%; Grade 4-occupying 75–100%. Brodsky’s classification [28] was used for tonsilar hypertrophy; Grade 0-tonsils within the tonsilar fossa, Grade 1-occupying < 25% of oropharyngeal width, Grade 2-occupying 26–50%, Grade 3-occupying 51–75%, Grade 4-occupying > 75%. After proper consent from parents or caregivers, OSA-18 QOL Questionnaire was recorded (Table 2).

Table 2.

OSA-18 quality of life questionnaire

	None of the time	Hardly any of the time	A little of the time	Some of the time	A good bit of the time	Most of the time	All of the time
1. Sleep disturbance
During the past 4 weeks, how often has your child had
Loud snoring	1	2	3	4	5	6	7
Breath-holding spells or pauses in breathing at night?	1	2	3	4	5	6	7
Choking or making gasping sounds while asleep?	1	2	3	4	5	6	7
Restless sleep or frequent awakening	1	2	3	4	5	6	7
2. Physical symptoms
During the past 4 weeks, how often has your child had
Mouth breathing because of nasal obstruction	1	2	3	4	5	6	7
Frequent colds or upper respiratory infections	1	2	3	4	5	6	7
Nasal discharge or runny nose	1	2	3	4	5	6	7
Difficulty swallowing	1	2	3	4	5	6	7
3. Emotional symptoms
During the past 4 weeks, how often has your child had
Mood swings or temper tantrums	1	2	3	4	5	6	7
Aggressive or hyperactive behavior	1	2	3	4	5	6	7
Discipline problems	1	2	3	4	5	6	7
4. Daytime function
During the past 4 weeks, how often has your child had
Excessive daytime sleepiness	1	2	3	4	5	6	7
Poor attention span or concentration	1	2	3	4	5	6	7
Difficulty getting up in the morning	1	2	3	4	5	6	7
5. Caregiver concerns
During the past 4 weeks, how often have the problems above
Caused you to worry about your child’s general health	1	2	3	4	5	6	7
Created concern that your child is not getting enough air	1	2	3	4	5	6	7
Interfered with your ability to perform daily activities	1	2	3	4	5	6	7
Made you frustrated	1	2	3	4	5	6	7

We have classified OSA-18 scores as; 18–36 normal, 37–60 mild, 61–80 moderate and 81–126 severe. Grades of adenoids and OSA-18 scores were used to individualize the treatment modalities and to evaluate their efficacy by comparing pretreatment and post-treatment OSA-18 score.

Patients were grouped into two groups depending upon OSA-18 scores and grades of adenoids.

Group 1—OSA-18 score < 80 and Adenoid hypertrophy < grade 3

Group 2—OSA-18 score > 81 and Adenoid hypertrophy > grade 3

Group 1 patients were treated with combination therapy of intranasal mometasone furoate spray (50 μg/puff) or fluticasone furoate spray (27.5 μg/puff) and syrup/tablet montelukast (5 mg) for 8 weeks whereas group 2 children were subjected to coblation assisted adenotonsillectomy under General anaesthesia. OSA-18 Questionnaire was recorded after 4 weeks of treatment. Post-treatment score of 18–36 or statistically significant difference indicated successful improvement and also the efficacy of treatment modality. For statistical analysis and to compare mean scores of each domain before and after intervention, Student’s paired t test was used. The values p < 0.05 were accepted as significant. The standardized response mean (SRM), defined as the difference score/SD of the difference score, was used to estimate the extent of improvement after surgery.

Observations and Results

The study enrolled 42 patients presented with clinical features of sleep disordered breathing, which were divided into 2 groups. Group A included 16 (38.1%) patients and group B had 26 (61.9%) patients. All patients were in age range of 3–15 years with mean of 6.67 years. 28 (66.67%) children were under the age of 3–8 years. Gender distribution showed male predominance with Male Female ratio of 2:1. Body mass index (BMI) varied from 12.2 to 26.6 kg/m² with mean of 16.45. Patients’ demographic data as per groups was illustrated in Table 3. In all cases, history and OSA-18 questionnaire were completed by parents, mostly mothers. 92.86% of parents share same bedroom with their children. Snoring and mouth breathing were most common complaints and present in all children. We inquired about nocturnal enuresis in all children and found that 17 (40.47%) children complained about nocturnal enuresis with frequencies > 3 nights per week. All 17 children OSA-18 score > 90 i.e. severe grade. 26 (61.9%) children had severe and 14 (33.33%) had moderate OSA-18 scores (Fig. 1). Grade 4 adenoid hypertrophy was seen in 52.38% children whereas Grade 3 and 2 were seen in 33.33% and 14.29% children respectively. Grade 3 and 4 tonsilar hypertrophy was observed in 38% and 28.57% children respectively. In Group B, 80.77% (21) and 57.69% (15) had grade 4 adenoid and tonsilar hypertrophy respectively. Group A showed grade 3 adenoid hypertrophy in 56.25% children and grade 2 in 43.75%.

Table 3.

Patients data as per group

Characteristic	Group A	Group B
Number	16	26
Gender (M/F)	10/6	18/8
M:F ratio	1.67:1	2.25:1
Age (years) (mean)	6.75 ± 2.46	6.62 ± 2.5
BMI (mean)	17.36 ± 3.83	15.88 ± 3.1

Fig. 1.

Showing distribution of patients as per OSA-18 score

In group A children, the OSA-18 domain with highest mean score was ‘physical symptoms’ followed by ‘caregivers concern’ and ‘sleep disturbances’. The mean pre-treatment and post-treatment OSA 18 scores were 70.31 and 33.5 respectively. Overall there was statistically significant improvement in QOL (p < 0.05) but 3 (18.75%) cases didn’t show much improvement. These children were subjected to surgical management. Post treatment improvement was highest for ‘physical symptoms’ and ‘sleep disturbances’ followed by ‘caregiver concerns’ (Table 4). Daytime problems and emotional symptoms showed lowest differences. In group B, ‘physical symptoms’ domain had highest score followed by ‘sleep disturbances’ and ‘caregiver concerns’. The mean preoperative score was 95.88 which improved to 24.92 after AT which was statistically significant (p < 0.05) (Table 5). As per our classification, all cases, after treatment, were in the class of 18–36 i.e. normal. All domains demonstrated significant improvement, maximum was seen in sleep disturbances and physical symptoms followed by caregiver concerns. While comparing total mean OSA-18 score and individual domain mean scores between group A and B, improvement was higher in group B which was statistically significant improvement.

Table 4.

OSA-18 score—pretreatment and post-treatment mean scores in Group 1

Domain	Pretreatment	Post-treatment	Difference
Sleep disturbance	14.69	6	8.69
Physical symptoms	18.94	7.2	11.74
Emotional symptoms	10.56	8.2	2.36
Daytime functions	9.1	5.6	3.5
Care givers concerns	15.38	6.3	9.1
Total	69.94	33.5	36.62

Table 5.

OSA-18 score—pretreatment and post-treatment mean scores in Group 2

Domain	Pretreatment	Post-treatment	Difference
Sleep disturbance	23.8	4	19.8
Physical symptoms	24	4.35	19.65
Emotional symptoms	14.5	8.1	6.4
Daytime functions	12	4.5	7.5
Care givers concerns	21.57	4.04	17.53
Total	95.88	24.92	70.96

Nocturnal enuresis All 17 cases were in the class of severe OSA-18 score and underwent adenotonsillectomy. In 11 (64.71%) children, nocturnal enuresis disappeared by 4th week and rest 6 children showed reduction in frequencies.

Discussion

Adenotonsillar hypertrophy is a most common cause of SDB in pediatric population. Numerous studies have shown potential links between OSA and related neurobehavioral and cardiopulmonary system morbidities [29–34]. These morbidities affect QOL of the children. PSG is the gold standard test and AHI is indicator of disease severity. Regarding candidacy of adenotonsillectomy, there are no specific clinical practice guidelines for preoperative PSG in pediatric OSA. Pediatric PSG testing should be reserved in cases; where clinical description didn’t match the clinical examination or medical comorbidities like obesity, craniofacial anomaly, syndromic children, mucopolysaccharidosis, sickle cell disease, neuromuscular diseases. Weatherly et al. [35], in their survey on practice patterns of otolaryngologists, found that less than 10% of patients received any objective testing and less than 5% of school-aged children received PSG prior to adenotonsillectomy. Actual practice pattern of otolaryngologists may reflect the fact that decisions or recommendation for management were based on clinical history as well as on physical examination demonstrating adenoid and/or tonsilar hypertrophy. However, predictive accuracy of clinically suspected SDB may be as low as 30% [36, 37]. Chervin RD et al. [38] demonstrated that if parental report of multiple measures of behavior is combined with medical history of SDB, the predictive accuracy of SDB becomes much higher.

Considering the higher costs and limited availability of pediatric PSG, alternative tools such as questionnaires were developed for quick evaluation and diagnosis. Questionnaires including symptoms-signs and consequences of SDB may help to identify severity of disease and hence the selection of appropriate management. A questionnaire which aids in diagnosis, treatment selection, measuring treatment outcomes and detection of residual SDB or symptoms that fail to improve may bridge the gap between objective tool like PSG and clinical suspicion. Example of such questionnaire is Pediatric Sleep Questionnaire (PSQ) developed by Chervin et al. [34] which demonstrated good validity and reliability.

In our study, we are using OSA-18 QOL questionnaire to aid clinical suspicion in early diagnosis of SDB and to determine its impact on QOL and thus the severity of disease. Franco et al. [20] showed significant relationship between OSA 18 scores and respiratory disturbance index. Kang et al. [24] had confirmed significant correlation between OSA-18 scores and PSG parameters, especially in domains of sleep disturbance, daytime function, caregiver concerns and total scores. We have classified OSA-18 questionnaire as; 18–36 normal, 37–60 mild, 61–80 moderate and 81–126 severe. The purpose of this classification was to categorize the severity of SDB, to select appropriate treatment modality and to evaluate efficacy of treatment in terms of improvement of QOL.

Role of adenotonsillectomy in treatment of pediatric OSA and its effect on QOL (using OSA-18) is well established by numerous studies [21–23]. Literature has documented effectiveness of medical management in mild cases of pediatric OSA [17, 19–21] but none of the studies have evaluated its effect on QOL by OSA-18 questionnaire. Using OSA-18, we have evaluated both QOL and effectiveness of medical management for mild SDB.

We observed male predominance with male to female ratio of 2:1, which was similar to various other studies in literature [9, 39, 40]. This finding is not consistent with other studies, which have shown that equal sex ratio [10, 21]. We noticed 66.67% children in the age range of 3–8 years. Epidemiological studies on pediatric SDB, have documented peak prevalence between 2 and 8 years [2, 5].

Out of 42 patients, we observed that 26 patients had adenoid and tonsils hypertrophy of grade 3 or 4 and 16 patients had less than 3. OSA-18 questionnaire score of patients with grade 3 or 4 adenotonsillar hypertrophy was more than 80 indicating severe impact of QOL whereas those with grade 3 or less had scores less than 80 indicating mild to moderate impact. We noticed that 80.77% (21) and 57.69% (15) children of group B had grade 4 adenoid and tonsilar hypertrophy respectively whereas Group A had 56.25% children grade 3 adenoid hypertrophy. These findings of our study positively supports relation between size of adenotonsillar hypertrophy and severity of impact of SDB [25, 26]. Thus OSA-18 score adenotonsillar hypertrophy can predict the severity of OSA.

In group A, higher mean scores were observed in domains of ‘physical symptoms’, ‘caregiver concern’, and ‘sleep disturbances’ where as in group B, higher scores were observed in ‘physical symptoms’, ‘sleep disturbances’ and ‘caregiver concern’. Many studies in literature reported same domains with higher score [9, 21, 23]. Snoring, mouth breathing, restless sleep and nasal symptoms were present in all our patients as a part of classical presentation of SDB, thus resulting in higher scores in physical symptoms and sleep disturbances. 92.86% parents have shared same bedroom with children, thus observation of sleep and respiratory patter disturbances become more obvious leading to raised anxiety amongst parents. This may be the reason for higher ‘caregivers concern’ scores. Kljajic et al. [41] in their study on Obstructive sleep apnea in children—how it affects parental psychological status, concluded that OSA in children is a disturbing symptom for parents and is associated with a significant level of anxiety which improved after treatment.

In both groups, post-treatment improvement in mean OSA-18 scores and hence in the impact on QOL was statistically significant. This finding reflects the efficacy of surgical management for severe SBD and medical management for mild moderate SDB. In group B, after treatment, highest improvement was noticed in ‘sleep disturbances’ domain followed by ‘physical symptoms’ and ‘caregivers concern’. Obvious improvement was observed in snoring and choking/breath holding spells after surgical clearance of airway obstructions, which contributes for higher improvement in ‘sleep disturbance’ domain. Group A, ‘physical symptom’ domain had highest improvement followed by ‘caregivers concern’ and ‘sleep disturbance’. Improvement in ‘physical symptom’ domain may be attributed to control of upper respiratory tract infections, allergic rhinitis and nasal discharge due to medical management.

For both groups, ‘emotional symptom’ domain showed significant improvement, but amongst all, it had least improvement which was similar to many international studies (after adenotonsillectomy) [9, 20, 38, 42]. This may be attributed to cultural differences, upbringing of child and other underlying behavioral conditions like ADHD. Authors like Silva et al. [21] and Begzada Hasukic [22] observed ‘daytime symptoms’ as a domain with least improvement.

In our study, 81.25% cases of mild to moderate SDB had improvement after medical therapy which was found to be higher when compared to other studies which reported improvement revolving around 40–70% [16, 18, 43, 44]. We found medical management as an effective and adequate treatment modality for mild to moderate SDB. Numerous studies agree with our result and support the fact “medical management may be a reasonable alternative to adenotonsillectomy for children with mild obstructive sleep apnea who are poor surgical candidates or whose parents desire to avoid surgical intervention” [15–18, 45].

We noticed nocturnal enuresis in 17 (40.47%) children who had severe OSA. After AT, 11 (64.71%) children showed complete resolution by 4 weeks. In study by Weissbach et al. [46], 27 children had nocturnal enuresis and all had severe OSA. Out of 27, 11 (41%) had complete resolution by 1st month and in 3 patients, it disappeared throughout the remaining time of follow-up. OSA-18 questionnaire doesn’t include nocturnal enuresis, but we considered it as a factor which significantly impacts the QOL of both child and parents. It also affects the social life of parents, contributing to caregiver’s anxiety and frustration. Thus enuresis needs special mention in SDB screening tool.

Conclusion

OSA-18 QOL questionnaire is quicker, self-administered and disease specific screening tool for early detection and quantification of impact of SDB on quality of life in pediatric patients. Being subjective tool, it can be an adjunct to clinical examination to improve the diagnosis of pediatric OSA and not a replacement of pediatric PSG. Along with grades of adenotonsillar hypertrophy, OSA-18 scores can be used to predict the severity of SDB and to categorize the patients for advocating appropriate treatment (Medical and Surgical) modalities. OSA-18 scores showed improvement in both groups after treatment. Thus we can conclude that medical management is effective for mild to moderate cases and surgical management for severe cases.

Funding

No funding sources.

Compliance with Ethical Standards

Conflict of interest

The authors declare that they have no conflict of interest.

Disclosure

This work has never been published and is not currently under evaluation in any other peer reviewed publication.

Ethical Approval

The study was approved by the Institutional Ethics Committee.

Informed Consent

Informed consent was obtained from parents’ caregivers all individual participants included in the study.