Study of quality of life outcome after adenotonsillectomy in children with sleep disorderd breathing

INTRODUCTION

Sleep-disordered breathing (SDB) is a common entity in children and includes a continuum of sleep-related breathing disturbances, SDB encompasses the spectrum of sleep disorders ranging in severity from primary snoring to obstructive sleep apnea(OSA). OSA on the severe end of the spectrum, and is generally diagnosed clinically based on signs and symptoms.¹ Classification of SDB.²

• • Primary snoring
• • Upper airway resistance syndrome (URAS)
• • Obstrutive sleep apnoea syndrome (OSAS)

Children with sleep disordered breathing (SDB) can manifest a continuum from simple snoring and upper airway resistance syndrome to obstructive sleep apnea (OSA).^3,4 The peak incidence of OSAS in children occurs between 2-8 years of age and parallels to the prominent growth of lymphoid tissue around the airway during these years however OSAS occurs in children of all ages, as early as neonatal period to the adolescent age group.⁵ Snoring and difficulty in breathing during sleep are the most common complaints of parents of children with OSAS.^6-8 Other symptoms include excessive daytime sleepiness, behavior problems, learning disabilities, right sided heart failure, growth retardation or failure to thrive.

Objective

The objective was to study the quality of life outcome after adenotonsillectomy in children with sleep disorderd breathing.

METHODS

This prospective study was conducted in the Department of Otorhinolaryngology Head and Neck Surgery, Government Medical College and associated SMHS Hospital Srinagar from July 2015 to July 2017. In this study we included the patients with SDB in pediatric age group, who visited in our OPD.

Diagnostic methods that have been scientifically evaluated include history and physical examination, audiotaping or videotaping, pulse oximetry, abbreviated polysomnography, and full polysomnography

• • History and physical evaluation.^9-11
• • Sleep history screening for snoring should be a part of routine health care visit. A more detailed history regarding labored breathing during sleep, observed apnea restless sleep, diaphoresis, anuresis, excessive daytime sleepiness and behaviour and learning problem (including attention deficit and hyperactivity disorder) should be obtained. On physical examination finding during wakefulness are often normal. There may be nonspecific finding related to adenotonsillar hypertrophy such as mouth breathing nasal obstruction during wakefulness, adenoid facies and hyponasal speech. Evidence of complication of OSAS may be present such as systemic hypertension and poor growth.
• • Video recording.¹²

A home based video recording of apneic episodes for 30 minutes can reliably be screened for OSAS in children.

• • Polysomnography in patients who cooperates.¹³

All the patients with adenotonsillar hyperplasia who fail to respond to conservative management were subjected to adeno-tonsillectomy surgery.

• • To assess the quality of life with OSAS before and after adenoidectomy or adenotonsillectomy OSA 18 questionnaires were used.¹⁴ The OSA 18 has 18 items in 5 domains including sleep disturbances, physical symptoms, emotional symptoms, daytime functions and caregiver concerns. The question was administered to the patient 2 weeks before and at 2 months and 6 months after surgery. There are total 18 questions and each question score between 1-7 and total score is 126. The impact on the quality of life is classified as mild<60, moderate 60-80 and severe >80. Dramatic improvement in the total score after T&A has been shown in number of studies within 6 months after surgery.

Inclusion criteria

Children age between 2-18 years with disorder of breathing during sleep characterized by prolonged partial upper airway obstruction or intermittent complete obstruction that disrupts normal ventilation during sleep and sleep patterns will be taken up for study. Children with following symptoms will also be evaluated for presence or otherwise of SDB.

• a) Snoring
• b) Mouth breathing
• c) Aggressive behavior and diurnal problems
• d) Difficulty in getting up in the morning
• e) Excessive sleep during the day.
• f) Altered craniofacial development
• g) Poor school performance and learning disabilities
• h) Growth retardation
• i) Children with hyperactive behaviour
• j) Children with enuresis

Exclusion criteria

Exclusion criteria for prior adenoidectomy and tonsillectomy include: neuromuscular disease, cleft palate, psychiatric condition/cognitive impairement.

Statistical analysis

Data was entered in a Microsoft excel spread sheet. Categorical variables were summarized as frequency and percentage. Continuous variables were summarized as mean and standard deviation. Paired samples t-test was used to compare OSA-18 score at pre-operative, 6 months and 1 year. Two-sided p-values were reported and a p<0.05 was considered statistically significant.

RESULTS

This prospective hospital based study was conducted in the Department of Otorhinolaryngology, Head and Neck Surgery, Government Medical College and Associated SMHS Hospital Srinagar. A total of 47 patients were included with following details.

Table 1 showing that our study group comprised of total 47 patients with majority of patients with in age group of 5-7 years. With youngest patient was 3 years and eldest was 15years old.

Table 2 showing that majority of patients in our study males were 29 (61.70%) and females were 18 (38.30%).

Table 3 showing that in our study the majority of patients in male group were within 5-7 years and while in female group the majority came also from 5-7 years.

Table 4 showing that in our study group of 47 patients the OSA 18 scale used pre-operative shows majority of patients 33 (70.21%) having score >80 score showing that there was a major impact on quality of life.

Table 5 indicates that there is significant improvement in OSA 18 score in postoperative period and hence improvement in quality of life.

Table 6 is showing that our study group of 47 patients mean OSA 18 Score pre operatively, at 2 months and at 6 months were 84.15, 30.4, 26.85 respectively. This signify that there is improvement in OSA-18 score and hence improvement in quality of life. P value was <0.001, which is statistically significant.

P value for OSA 18 pre op vs. OSA 18 at 2 months <0.001 (repeated measures ANOVA, Bonferroni post-hoc). P value for OSA 18 at 2 months vs. OSA 18 at 6 months <0.001 (repeated measures ANOVA, Bonferroni post-hoc)

Figure 1 is showing that there were positive correlation between PSG (AHI) score and OSA 18 score with coefficient of determination=0.778. (The dots represent the number of patients and the line defines the linear trend).

DISCUSSION

Sleep-disordered breathing (SDB) represents a spectrum of sleep-related diseases resulting in nocturnal breathing difficulty, ranging from intermittent and habitual snoring (HS) to obstructive sleep apnea syndrome (OSAS).SDB is extremely common; estimates suggest that HS and OSAS occur in 10% and 2% to 4% of children, respectively.¹⁵ Untreated pediatric SDB is associated with numerous comorbidities that include lowered IQ, decreased executive function, behavioural problems, secondary nocturnal enuresis, and cardiopulmonary dysfunction.¹⁶

In the present study, we had 47 patients with majority of patients were males 29 (61.70%) and females were 18 (38.30%), with majority of patients were in the age group of 5-7 yrs (48.94%) (Table 1 to 3). Jose mario de lima jr et al also found in their study that 56.2% were males and 43.8% were females with mean age of 6 yrs and 6 months.¹⁷ Ron et al also found in their study that 51% were males and 49% were females with mean age of 6.3 yrs i.e. the male preponderance.¹⁸ Valerie et al found in their study that 53% were females and 47% were males with mean age of 6 yrs.¹⁹ It is a consensus among various authors that adenoid or adenotonsillary hyperplasia is the main cause of SDB among children.^20-23 Adenoidectomy (or adenotonsillectomy) have been, therefore, procedures of choice for the treatment of most cases of SDB in the pediatric population. These procedures have shown positive results in reverting the physical effects of SDB.^24-27 Adenoidectomy/adenotonsillectomy not only revert the organic changes caused by SDB, but also have shown their value in improving the quality of life of children and the perception of this quality by their caretakers. Recent papers are evidence of this important theme.^28-33 In the present study of 47 patients 41 (87.23%), 4 (8.51%) underwent adenotonsillectomy and tonsillectomy respectively.

PSG (polysomnography) is recognized as the gold standard for establishing a diagnosis of OSA, constraints such as its limited availability and expense have left many clinicians searching for more and more widely available and less costly diagnostic tool.³⁴ Numerous studies investigating the validity of using history and physical examination alone to establish an OSA diagnosis have found that they are poor predictors of OSA.^35-42 Additional studies have evaluated the usefulness of questionnaires to identify OSA. One such questionnaire is the OSA-18, which is a disease-specific quality-of-life questionnaire commonly used by pediatric otolaryngologists.⁴³ Because this survey includes a number of symptom-specific questions and was validated against 90-minute, daytime-nap sleep studies, two investigations examined its utility as a potential diagnostic tool. The OSA-18 questionnaire, developed by Franco Jr. et al was applied in three moments to the caretakers of children including in this study to assess the quality of life of these patients.²⁸ In the first of these moments - a preoperative - a baseline score was obtained as a parameter for assessing the quality of life of children before adenoidectomy or adenotonsillectomy. According to Franco Jr et al, patients scoring below 60 experience low impact on their quality of life.²⁸ Patients scoring between 60 and 79 are moderately affected; if the score is 80 points or over, there is major impact on the quality of life. The OSA-18 questionnaire was applied at 2 months and at 6 months after surgery; the aim was to assess the long-term effects on the quality of life of children.

In the present study of 47 patients OSA-18 questionnaire was used pre-operatively, at 2 months, at 6 months. OSA-18 questionnaire when used pre-operatively showed majority of patients 33 (70.21%) having score >80, and 14 (29.79%) patients having score between 60-80. No patient was below 60 score. Mean OSA-18 score pre-operatively was 84.15 Table 4 and 6. OSA-18 score at 2 months and at 6 months were 30.06 and 26.40 respectively, which showed a lot of improvement in postoperative score and hence the post –operative quality of life Table 5. De Lima Junior et al in their prospective study of 48 patients found that before surgery, the average OSA-18 score was 82.83 (SD=12.57).¹⁷ Within thirty days after surgery, the average OSA-18 score was 34.3 (SD=9.95), both showing significant reduction (p<0.001). Thus they concluded that surgery improves quality of life of children with OSD, which is consistent with the results of our study. Valerie et al in their study used OSA-18 questionnaire pre and post –operatively.¹⁹ OSA-18 scores showed improvement post-operativly in all domains, which is consistent with our study. Franco et al in their study also found that there was a great improvement in quality of life in patients undergoing adenotonsillectomy, which is consistent with our study.²⁸ de Serres et al performed a multi-institutional study to evaluate the quality of life in children undergoing adenotonsillectomy using OSA-6 which showed improvement in the quality of life.³⁰

CONCLUSION

Based on the observation made in the study we concluded that there was a significant improvement in the disease specific postoperative quality of life after both adenotonsillectomy and tonsillectomy. We also concluded that polysomnography results matched with OSA 18 questionnaires scale score done preoperatively (Figure 1).

Funding: No funding sources

Conflict of interest: None declared

Ethical approval: The study was approved by the Institutional Ethics Committee

REFERENCES

1. 1. Guilleminault C, Pelayo R, Leger D, Clerk A, Bocian RC. Recognition of sleep-disordered breathing in children. Pediatrics. 1996;98:871–82.
2. 2. Coleman JA. Pathophysiology of snoring and obstructive sleep apnea: airway dynamics. In: Fairbanks DNK, Mickelson SA, Woodson BT, eds. Snoring and Obstructive Sleep Apnea. 3rd ed. Philadelphia: Williams & Wilkins Co.; 2003: 19.
3. 3. Ali NJ, Pitson DJ. Snoring, Sleep disturbance, and behavior in 4-5 year olds. Arch Dis Child. 1993;68:360-6.
4. 4. Redline S, Tishler PV, Schluchter M, Aylor J, Clark K, Graham G. Risk factors for sleep disordered breathing in children: associations with obesity, race, and respiratory problems. Am J Respir Crit Care Med. 1999;159:1527-32.
5. 5. American Thoracic Society. Standard and indications for Cardiopulmonary sleep studies in children. Am J Respir Crit Care Med. 1996;153:866-78.
6. 6. Arens R, Marcus CL. Pathophysiology of upper airway obstruction:a developmental perspective. Sleep. 2004;27:997-1019.
7. 7. Guilleminault C, Korobkin R, Winkle R. A review of 50 children with obstructive sleep apnea syndrome. Lung. 1981;159:275–87.
8. 8. Brouilette R, Hanson D, David R, Klemka L, Szatkowski A, Fernbach S, Hunt C. A diagnostic approach to suspected obstructive sleep apnea in children. J Pediatr. 1984;105:10–14.
9. 9. Durr ML, Meyer AK, Kezirian EJ, Mamlouk MD, Frieden IJ, Rosbe KW. Sleep-Disordered Breathing in Pediatric Head and Neck Vascular Malformations. Laryngoscope. 2017;127(9):2159-64.
10. 10. Guilleminault C, Korobkin R, Winkle R. A review of 50 children with obstructive sleep apnea syndrome. Lung. 1981;159:275–87.
11. 11. Brouilette R, Hanson D, David R, Klemka L, Szatkowski A, Fernbach S, et al. A diagnostic approach to suspected obstructive sleep apnea in children. J Pediatr. 1984;105:10–4.
12. 12. Spruyt K, Gozal D. Pediatric sleep questionnaires as diagnostic or epidemiological tools: A review of currently available instruments. Sleep Medicine Reviews. 2011;15(1):19-32.
13. 13. Shouldice RB, O’Brien LM, O’Brien C, de Chazal P, Gozal D, Heneghan C. Detection of obstructive sleep apnea in pediatric subjects using surface lead electrocardiogram features. Sleep. 2004;27:784–92.
14. 14. Section on Pediatric Pulmonology, Subcommittee on Obstructive Sleep Apnea Syndrome. American Academy of Pediatrics. Clinical practice guideline: diagnosis and management of childhood obstructive sleep apnea syndrome. Pediatrics. 2002;109:704–12.
15. 15. Goldbart AD, Tal A. Inflammation and sleep disordered breathing in children:a state-of-the-art review. Pediatr Pulmonol. 2008;43:1151–60.
16. 16. Cardiorespiratory sleep studies in children. Establishment of normative data and polysomnographic predictors of morbidity, American Thoracic Society. Am J Respir Crit Care Med. 1999;160:1381.
17. 17. de Lima Jr JM, da Silva VC, de Freitas MR. Long term results in the life quality of children with obstructive sleep disorders Rev Bras Otorrinolaringol. 2008;74(5):718-24.
18. 18. Ron B. Kelly MJ. Outcomes and Quality of life following Adenotonsillectomy for sleep-dis0rdered breathing in children. ORL. 2007;69:345-8.
19. 19. Flanary VA. Long-Term Effect of Adenotonsillectomy on Quality of Life in Pediatric Patients. Laryngoscope. 2003;113:1639-44.
20. 20. Leach J, Olson J, Hermann J, Manning S. Polysomnographic and clinical findings in children with obstructive sleep apnea. Arch Otolaryngol Head Neck Surg. 1992;118(7):741-4.
21. 21. Reilly JS. Apnéia obstrutiva do sono e ronco em crianças: noções gerais. Em: Sih T, Chinski A, Eavey R, editores. III Manual de Otorrinolaringologia Pediátrica da IAPO; 2003: 59-66.
22. 22. Messner AH, Pelayo R. Pediatric sleep-related breathing disorders. Am J Otolaryngol. 2000;21(2):98-107.
23. 23. Weckx LLM, Weckx LY. Respirador bucal: causas e conseqüências. Rev Bras Med. 1995;52(8):863-74.
24. 24. Brooks LJ. Treatment of otherwise normal children with obstructive sleep apnea. Ear Nose Throat J. 1993;72(1):77-9.
25. 25. Potsic WP, Pasquariello PS, Baranak CC, Marsh RR, Miller LM. Relief of upper airway obstruction by adenotonsillectomy. Otolaryngol Head Neck Surg. 1986;94(4):476-80.
26. 26. Shintani T, Asakura K, Kataura A. The effect of adenotonsillectomy in children with OSA. Int J Pediatr Otorhinolaringol. 1998;44(1):51-8.
27. 27. Stradling JR, Thomas G, Warley AR, Williams P, Freeland A. Effect of adenotonsillectomy on nocturnal hypoxaemia, sleep disturbance and symptoms in snoring children. Lancet. 1990;335(8684):249-53.
28. 28. Franco RA Jr, Rosenfeld RM, Rao M. First place resident clinical science award 1999. Quality of life for children with obstructive sleep apnea. Otolaryngol Head Neck Surg. 2000;123(1):9-16.
29. 29. Stewart MG, Friedman EM, Sulek M, Hulka GF, Kuppersmith RB, Harrill WC, et al. Quality of life and health status in pediatric tonsil and adenoid disease. Arch Otolaryngol Head Neck Surg. 2000;126(1):458.
30. 30. De Serres LM, Derkay C, Astley S, Deyo RA, Rosenfeld RM, Gates GA. Measuring quality of life in children with obstructive sleep disorders. Arch Otolaryngol Head Neck Surg. 2000;126(12):1423-9.
31. 31. De Serres LM, Derkay C, Sie K, Biavati M, Jones J, Tunkel D, et al. Impact of adenotonsillectomy on quality of life in children with obstructive sleep disorders. Arch Otolaryngology Head Neck Surg. 2002;128(5):489-96.
32. 32. Goldstein NA, Fatima M, Campbell TF, Rosenfeld RM. Child behavior and quality of life before and after tonsillectomy and adenoidectomy. Arch Otolaryngol Head Neck Surg. 2002;128(7):770-5.
33. 33. Sohn H, Rosenfeld RM. Evaluation of sleep-disordered breathing in children. Otolaryngol Head Neck Surg. 2003;128(3):344-52.
34. 34. Ishman SL. The role of sleep studies in children who snore. JAMA Otolaryngol Head Neck Surg. 2016;142(2):179-81.
35. 35. Bixler EO, Vgontzas AN, Lin HM, Liao D, Calhoun S, Vela-Bueno A, et al. Sleep-disordered breathing in children in a general population sample: prevalence and risk factors. Sleep. 2009;32:731–6.
36. 36. Schecter MS. American Academy of Pediatrics, Section on Pediatric Pulmonology, Subcommittee on Obstructive Sleep Apnea Syndrome. Technical report: diagnosis and management of childhood obstructive sleep apnea syndrome. Pediatrics. 2002;109:69–88.
37. 37. Brietzke SE, Katz ES, Roberson DW. Can history and physical examination reliably diagnose pediatric obstructive sleep apnea/hypopnea syndrome? A systematic review of the literature. Otolaryngol Head Neck Surg. 2004;131:827–32.
38. 38. Carroll JL, McColley SA, Marcus CL, Curtis S, Loughlin GM. Inability of clinical history to distinguish primary snoring from obstructive sleep apnea syndrome in children. Chest. 1995;108:610–8.
39. 39. Goldstein NA, Sculerati N, Walsleben JA, Bhatia N, Friedman DM, Rapoport DM. Clinical diagnosis of pediatric obstructive sleep apnea validated by polysomnography. Otolaryngol Head Neck Surg. 1994;111:611–7.
40. 40. Leach J, Olson J, Hermann J, Manning S. Polysomnographic and clinical findings in children with obstructive sleep apnea. Arch Otolaryngol Head Neck Surg. 1992;118:741–4.
41. 41. Suen JS, Arnold, JE, Brooks LJ. Adenotonsillectomy for treatment of obstructive sleep apnea in children. Arch Otolaryngol Head Neck Surg. 1995;121:525–30.
42. 42. Wang RC, Elkins TP, Keech D. Wauquier A, Hubbard D. Accuracy of clinical evaluation in pediatric obstructive sleep apnea. Otolaryngol Head Neck Surg. 1998;118:69–73.
43. 43. Franco RA, Rosenfeld RM, Rao M. Quality-of-life for children with obstructive sleep apnea. Otolaryngol Head Neck Surg. 2000;123:9–16.