Quality of Life and Behavioural Assessment in Post-adenotonsillectomy Cases of Paediatric Age Group

Abstract

Adenotonsillectomy is a widely practiced surgical intervention to manage obstructive sleep apnoea syndrome (OSAS) in the paediatric age group. We conducted a prospective, non randomised, interventional study among 40 patients in paediatric age group (2–18 years) suffering from adenotonsillar hypertrophy with an indication of adenotonsillectomy based on American Academy of Otolaryngology-Head and Neck Surgery criteria. Care givers completed the obstructive sleep apnoea 18 (OSA 18) quality of life (QoL) survey and Rutter Children’s Behaviour Questionnaire (RCBQ) before adenotonsillectomy as well as 6 months after surgery. We found that mean score of OSA 18 and total RCBQ decreased significantly 6 months after surgery. The mean score of OSA 18 (4.12 ± 1.4) and total RCBQ score (20.5 ± 2) significantly reduced to 1.19 ± 0.12 and 7.4 ± 2.55 (p < 0.0001), respectively, after surgery. There was significant improvement in all the domains of OSA 18 score after intervention including sleep disturbance (mean score change 2.56, p < 0.0001), physical suffering (2.99, p < 0.0001), emotional distress (2.71, p < 0.0001), daytime problems (2.56, p < 0.0001), caregiver concerns (3.76, p < 0.0001). We also observed that QoL as well as behaviour was independent of age and gender of patients. This study demonstrates significant score changes across all questionnaire domains, comparing pre and post operative data indicating an improvement in their quality of life and behaviour. Hence, we advocate that adenotonsillectomy may be considered as an effective treatment in properly selected patients with OSAS.

Introduction

Adenotonsillar hypertrophy is one of the most common problems in the ENT practices. It is responsible for severe impact on child development and behaviour, which is a major cause of concern among guardians and physicians. The most common cause of chronic airway obstruction in children is adenoid hypertrophy, which exhibits variety of clinical signs and symptoms, collectively termed as Obstructive sleep apnoea syndrome (OSAS), which affects 1–2% of paediatric population [1]. Clinical features of OSA include mouth breathing, snoring, restless sleep, choking, gasping, unusual sleeping positions frequent awakenings, morning lethargy and daily fatigue. It can cause more serious complications including cardiovascular, neurobehavioral and growth disorders [2, 3]. Neurobehavioral problems include cognitive and memory impairment, increased daytime sleepiness, emotional discomfort as irritability and impatience, and learning deficit. The impact of Sleep Disordered Breathing (SDB) on childhood development and behaviour, specially hyperactivity and inattention have been well published [4–7]. The Eustachian tube may become blocked due to hypertrophic adenoids, potentially leading to recurrent otitis media which may aggravate attention deficit and poor academic performances. Pulmonary hypertension and Cor pulmonale are less common but more severe occurrences. They are considered as long term alterations of adenotonsillar hypertrophy [8, 9]. Hence, there has been increasing interest in the measurement of the quality of life (QoL) of children with OSA, in order to rectify these problems. The target of this study is to make a comparison between pre and postoperative signs and symptoms of selected patients undergone adenotonsillectomy, using tools which measures patient’s QoL and behaviour. This is the first Indian study which not only used a disease specific quality of life tool (OSA 18 QoL survey) to assess children affected by features of adenotonsillar hypertrophy but also included an additional questionnaire (RCBQ) to assess the behavioural changes following adenotonsillectomy. It was eventually hypothesised that the QoL and behaviour of children, as measured by a validated QoL and behaviour questionnaire, improves following adenotonsillectomy.

Materials and Methods

We conducted the study in VMMC and Safdarjung Hospital, New Delhi, in the department of ENT in 40 patients aged between 2 and 18 years over the period of 18 months. Patients suffering from adenotonsillar hypertrophy with an indication of adenotonsillectomy based on American Academy of Otolaryngology-Head and Neck surgery (AAO-HNS) criteria and sleep as well as behaviour disorder were included in the study. We designed this as a prospective, non-randomized, interventional study.

All the patients were pre-operatively evaluated clinically as well as radiologically. A uniform history was documented for every patient and necessary routine investigations were done. OSA-18 (Obstructive Sleep Apnoea-18) Quality of Life Survey Questionnaire and parental Rutter Children’s Behaviour Questionnaire (RCBQ) were applied for the assessment (“Appendices 1, 2” section). Patient’s parents or caregivers were explained the questionnaire method and asked to fill the same before operation as well as 6 months after operation.

OSA-18 questionnaire is the most widely used tool to measure the Quality of Life of children with sleep disordered breathing. It consists of 18 survey items divided into five domains, each with 3–4 individual items which are as follows

• Sleep disturbance,

• Physical suffering,

• Emotional distress,

• Daytime problems and

• Caregiver concerns.

Each of 18 items gives a score from 1 (lowest) to 7 (highest). The mean score for each domain can be obtained by averaging the scores of the items within the domain. The overall mean score is obtained by averaging the 18 items. OSA-18 has been psychometrically validated and shown to be reliable and responsive [10].

The RCBQ, an 18-item questionnaire which includes description of the child’s behaviour is asked to be filled by parents or caregivers. Parents indicate whether each description ‘does not apply’, ‘applies somewhat’, or ‘Certainly applies’. RCBQ scoring is performed by summing responses given to each item from 0 to 2, where a score of 0 indicates ‘Does not apply’; 1, ‘Applies somewhat’ and 2, ‘Certainly applies’. RCBQ result is considered “normal” if the score is below the 80th percentile; “moderate problem” if the score is between the 80th and 95th percentile; and “severe problem” if the score is above the 95th percentile. RCBQ has been used in various studies worldwide and its validity and reliability has been demonstrated previously [11].

Results

All of the domains and the overall score in OSA-18 Questionnaire showed a large degree of improvement. We calculated the statistical significance of the score using the Wilcoxon signed rank test. The effect size or the standardized response mean was calculated by dividing the score change by its standard deviation. An effect size of greater than 0.8 was considered large. By this definition, the calculated effect sizes for all the domains and scores pertaining to OSA-18 were large. The largest effect sizes were for caregiver concerns and physical sufferings while the smallest were for day time problem and sleep disturbance.

Mean sleep disturbance score, physical suffering score, emotional distress score, daytime problem score, caregiver concern score and overall mean score of OSA-18 pre-intervention was 3.74 ± 1.7, 4.24 ± 1.47, 3.95 ± 1.6, 3.7 ± 1.89, 4.91 ± 1.61 and 4.12 ± 1.4, respectively whereas post-intervention was 1.18 ± 0.24, 1.24 ± 0.21, 1.24 ± 0.25, 1.14 ± 0.17, 1.14 ± 0.19 and 1.19 ± 0.12, respectively. Mean overall scores and domains was significantly lower after intervention as compared to before intervention (p < 0.0001).

The effect sizes (95% Confidence Interval) for the domain of sleep disturbance, physical suffering, emotional distress, daytime problem, caregiver concern and overall mean score of OSA-18 was found to be 1.45 (1.43–1.96), 2.03 (2.21–2.91), 1.67 (1.21–1.95), 1.36 (1.17–1.72), 2.35 (1.55–2.11), 2.09 (1.43–2.23), respectively (Table 1; Fig. 1).

Table 1.

Comparison of OSA 18 mean scores and its domain before and after intervention

Domain	Before			After			Mean score change ± SD	Effect size (95% CI)	p Value
	Mean ± SD	Median	Min–max	Mean ± SD	Median	Min–max
Sleep disturbance	3.74 ± 1.7	4.38	1–6	1.18 ± 0.24	1	1–1.75	2.56 ± 1.77	1.45 (1.43–1.96)	< .0001
Physical suffering	4.24 ± 1.47	4.62	1–6	1.24 ± 0.21	1.25	1–1.75	2.99 ± 1.47	2.03 (2.21–2.91)	< .0001
Emotional distress	3.95 ± 1.6	4.34	1–6.38	1.24 ± 0.25	1.33	1–1.67	2.71 ± 1.62	1.67 (1.21–1.95)	< .0001
Daytime problems	3.7 ± 1.89	3.67	1–7	1.14 ± 0.17	1	1–1.33	2.56 ± 1.88	1.36 (1.17–1.72)	< .0001
Caregiver concerns	4.91 ± 1.61	5.5	1–7	1.14 ± 0.19	1	1–1.5	3.76 ± 1.6	2.35 (1.55–2.11)	< .0001
Mean score	4.12 ± 1.4	4.25	1.17–6.17	1.19 ± 0.12	1.19	1–1.33	2.93 ± 1.4	2.09 (1.43–2.23)	< .0001

Fig. 1.

Comparison of OSA score before and after adenotonsillectomy

We compare the total OSA-18 score and total RCBQ score before and after intervention. Mean total score of OSA-18 and RCBQ before intervention was 74.22 ± 25.14 and 20.5 ± 2, respectively which was significantly reduced to 21.42 ± 2.12 and 13.1 ± 1.6, respectively (p < 0.0001),concluding that after intervention total OSA-18 and RCBQ scores improved significantly (Table 2; Fig. 2).

Table 2.

Comparison of OSA18 and RCBQ total scores before and after intervention

Total score	Before			After			Mean score change ± SD	p Value
	Mean ± SD	Median	Min–max	Mean ± SD	Median	Min–max
Total score OSA 18	74.22 ± 25.14	76.5	21–111	21.42 ± 2.12	21.5	18–24	52.8 ± 25.19	< .0001
Total score RCBQ	20.5 ± 2	20	18–27	13.1 ± 1.61	14	11–16	7.4 ± 2.55	< .0001

Fig. 2.

Comparison of total score OSA and total score RCBQ before and after adenotonsillectomy

77.50% of study population had normal RCBQ score severity level before adenotonsillectomy which increased after adenotonsillectomy to 80%. 17.50% of patients had moderate problem before intervention which decreased to 15% after intervention, while 5% of patients persisted with severe problem. Thus it can be concluded that there was no statistically significant change in before and after intervention severity levels of RCBQ score (p = 0.955) (Table 3, Fig. 3).

Table 3.

Change in severity levels of RCBQ before and after intervention

	Before intervention RCBQ	After intervention RCBQ	p Value
Normal	31 (77.50%)	32 (80.00%)	0.955
Moderate problem	7 (17.50%)	6 (15.00%)
Severe problem	2 (5.00%)	2 (5.00%)
Total	40 (100.00%)	40 (100.00%)

Fig. 3.

Change in severity levels of RCBQ before and after intervention adenotonsillectomy

We assessed correlation of age and sex with sleep disturbance, physical suffering, emotional distress, daytime problems, caregiver’s concern, mean and total score of OSA-18 and total score of RCBQ using Spearman rank correlation coefficient. We did not find any correlation between the variables.

Discussion

The prevalence of obstructive sleep apnoea syndrome (OSAS) is 0.7–1.8% [12]. Obstructive sleep apnoea is defined as a ‘disorder of breathing during sleep characterized by prolonged partial upper airway obstruction and/or intermittent complete obstruction that disrupts ventilation during sleep and normal sleep pattern’ [12]. Primary snoring is defined as noisy breathing without obstructive apnoea, frequent arousal from sleep or gas exchange abnormalities. Sleep disordered breathing is used to describe both snoring and OSA [12]. Such patients complain about snoring, mouth breathing, gasping, apnoea, enuresis, day time somnolence and behavioural changes. There may be serious effects on behaviour including poor school performance and aggression. The neurobehavioural consequences of paediatric OSAS are well established in literature. Studies have shown that children having OSAS suffer from poor cognitive function and memory. The quality of life also gets affected by OSAS. Cardiovascular complications like cor pulmonale, arrhythmia and hypertension are notable serious concerns of physician while treating patients of OSAS.

The largest group of children who do not have underlying medical diagnosis suffer from physical obstruction of upper airway due to adenotonsillar hypertrophy. Hence, adenotonsillectomy is one of the common surgeries performed to treat paediatric OSAS.

The largest trial of OSAS is the Childhood Adenotonsillectomy Trial (CHAT) which assessed primary outcome as neuropsychological testing and secondary outcome as quality of life assessment between control arm and treatment arm [12]. There was no significant difference in neuropsychological outcome but quality of life showed a significant improvement among treatment arm in comparison with control arm. In contrast to this study, a study by Li et al. [13] showed no significant improvement in sleep apnoea after adenotonsillectomy, with no remarkable improvement in quality of life. This study used polysomnography (PSG), tests of variables of attention (TOVAs), and Child Behaviour Checklists to compare findings before and 6 months after surgery.

Wei et al. [14] used the Pediatric Sleep Questionnaire (PSQ) and Conners’ Parent Rating Scale-Revised Short Form (CPRS-RS) in 117 children with SDB who underwent adenotonsillectomy. It was completed before and 6 months after surgery. It concluded that both sleep and behaviour improved 6 months after adenotonsillectomy.

Valerie A. Flanary, Mitchell et al. and Powell et al. used the OSA-18 quality of life questionnaire and concluded that adenotonsillectomy markedly improves quality of life in children with either obstructive sleep apnoea or mild sleep disordered breathing [15]. In all of these studies though all the domains showed improvement in score in the post operative period, the highest improvement was observed in sleep disturbance score.

OSA-18 quality of life survey is an instrument developed and validated by Rosenfeld et al. and has been widely used in many previous studies [10]. Similarly RCBQ is a previously validated clinometric instrument used for evaluation of a child’s behavioural changes [11].

We assessed the impact of adenotonsillectomy on the QoL and behaviour of patients aged between 2 and 18 years. We found that OSA-18 scores showed improvement in all domains after 6 month follow up period. Domains of greatest improvement include caregiver concerns and physical sufferings while least improvement were for day time problem and sleep disturbance. Overall OSA-18 mean score change was 2.93 (p < 0.0001) corresponding to an overall QOL improvement. Total RCBQ mean score change was significant (p < 0.0001), indicating an overall improvement in behaviour following adenotonsillectomy. However there was no statistically significant change in severity level of RCBQ score.

The mean OSA-18 questionnaire improvement scores reflect improvements in the child’s general QoL. Thus, our results indicate majority of patients (82.50%) had large change in score whereas only 7.50% of patients had trivial change, 7.5% patients had small change and 2.5% patients had moderate change. Thus, all the children’s QoL improved following adenotonsillectomy.

There was significant improvement in all the domains of OSA 18 score and in its mean overall score after intervention as compared to before intervention i.e. sleep disturbance (mean score change 2.56, p < 0.0001), physical suffering (2.99, p < 0.0001), emotional distress (2.71, p < 0.0001), daytime problems (2.56, p < 0.0001), caregiver concerns (3.76, p < 0.0001) and overall mean score (2.93, p < 0.0001). There was no significant association of change in overall mean score of OSA-18 with gender and age. Similarly there also was no statistically significant correlation of change in total score of RCBQ with age or gender. After intervention 80.00% of patients were with normal RCBQ score, 15.00% of patients were with moderate problem and 5% of patients persisted with severe problem.

There exists valid controversy in the literature about improvement of QoL after adenotonsillectomy. This surgical intervention undoubtedly removes the physical obstruction of upper airway in children suffering from OSAS. Significant reduction of central apnoea has been observed after adenotonsillectomy. Improvement of cognitive function has also been documented but some studies contradict the hypothesis. According to Bruno Giordani et al. [16] there was no significant improvement of OSAS or cognitive function after adenotonsillectomy. Caregiver’s assessment of quality of life improvement was biased after surgery. Reduction of hyperactive behaviour resulting from improved frontal cortical function after restoration of normal sleep could influence the observations of caregiver. Decline of cognitive function may be attributed to the possibility of central nervous system damage due to OSAS at early age [16].

We did not find any quality of life or neuropsychological deterioration after adenotonsillectomy. OSAS being multifactorial disease, selection of cases for adenotonsillectomy holds paramount importance prior to surgery. Incomplete removal of adenoid and tonsil, associated other clinical features like Down’s syndrome, craniofacial anomalies, obesity, unfavourable social surrounding may be hypothesised for unexpected outcomes following adenotonsillectomy.

The main limitations and weaknesses of this study include the lack of a control group, absence of the application of the full RCBQ scale A, and use of a subjective questionnaire for assessment of the primary outcome measure. PSG was not used to document the presence of upper airway obstruction (UAO) or OSA. To document the diagnosis of OSA and UAO, PSG is often used and is considered the gold standard for evaluation of these syndromes. This study relied exclusively on clinical examination for the diagnosis of upper airway obstruction and OSA. The accuracy of clinical examination may been questioned. However, on the basis of the clinical diagnosis and treatment, quality of life did improve in individuals who underwent adenotonsillectomy. We applied a new combination of survey (OSA 18 questionnaire and RCBQ) together, that has previously never been used.

Further cohorts, including control groups and larger sample sizes and longer follow up are warranted to replicate and confirm our results.

Conclusion and Recommendations

The OSA-18 questionnaire and RCBQ can easily be incorporated into clinical practice. We found that adenotonsillectomy causes a significant improvement in children’s quality of life and behaviour. We did not find any post-operative life threatening complications, infections, haemorrohage needing blood transfusion or re-evaluation in operation theatre following surgery. So, we recommend that adenotonsillectomy is an effective treatment in children suffering from OSAS in properly selected patients considering associated comorbidities, without any intra or post-operative complications.

Appendix 1

Appendix 2

Compliance with Ethical Standards

Conflict of interest

The authors declare that they have no conflict of interest.

Ethical Approval

All procedures involving human participants were in accordance with the ethical standards of the institution.

Informed Consent

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