AM ER IC AN JOURNAL OF OT OLA RYNGOLOGY–H E A D A N D NE CK M E D ICI N E AN D S U RGE RY 3 7 (2 0 1 6) 1 28 –1 3 1
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Pediatric otolaryngology: principles and practice
Evaluation of growth curves in children after supraglottoplasty☆,☆☆,★ John Neiner, MD⁎, Anil Gungor, MD LSU Health Shreveport Department of Otolaryngology, 1501 Kings Highway, Shreveport, LA
ARTI CLE I NFO
A BS TRACT
Article history:
Objectives: To determine if monitoring weight growth curves is a sensitive objective
Received 2 November 2015
parameter for evaluating operative outcomes after supraglottoplasty. Study design: Retrospective chart review. Methods: An IRB approved retrospective review of patients who underwent supraglottoplasty from 2/28/2012 to 10/20/2014 by the otolaryngology department at a single institution was performed. Variables collected included age, race, sex, preoperative weight percentiles, and weight percentiles at 3 month, 12 month, and 3 year followup intervals. Results: 20 patients met inclusion criteria. 15 (75%) patients were male and 5 (25%) were female. 9 (45%) patients were African American, 8 (40%) were Caucasian, and 3 (15%) were other. Average weight for age at surgery was 29.8 percentile. 6 (30%) had failure to thrive by weight. By 3 months postop average weight had increased by 7.67 percentile (p = 0.09, 95% CI − 1.62 to 17.0), by 12 months there was an observed increase of 19.1 percentile (p = 0.06, 95% CI 0.47–37.8), and by 3 years the average weight had increased by 26.53 percentile (p = 0.03, 95% CI 4.47-48.59). By three years postop the average weight had normalized (64.5 percentile). Among those who met preoperative failure to thrive criteria (average 0.11 percentile), weight gain was still dramatic with average weight percentile of 37.5 by 3 years postop. Conclusion: Patients undergoing supraglottoplasty are typically underweight for age. Statistically significant weight gain occurs in children after going supraglottoplasty. This intervention can normalize their growth chart growth patterns by 3 years postoperatively, even in children with failure to thrive. Published by Elsevier Inc.
1.
Introduction
A disorder of up to 10% of pediatric patients seen in the primary care setting, failure-to-thrive is typically characterized by weight for age that falls below the 5th percentile or significant weight deceleration resulting from an imbalance ☆
of caloric intake, absorption, and expenditure [1]. Poor weight gain has previously been reported in children with obstructive sleep apnea due to adenotonsillar hypertrophy with significant weight gain occurring after tonsillectomy, even among children classified preoperatively as failure-to-thrive. It is postulated that respiratory distress during feeding, increased
Each of the authors (Drs. Neiner and Gungor) has contributed to, read and approved this manuscript. This manuscript is original and it, or any part of it, has not been previously published; nor is it under consideration for publication elsewhere. ★ A Copyright Transfer Statement has been signed by all authors. ⁎ Corresponding author at: LSU Health Shreveport Department of Otolaryngology, 1501 Kings Highway, Shreveport, LA 71103. E-mail address:
[email protected] (J. Neiner). ☆☆
http://dx.doi.org/10.1016/j.amjoto.2015.11.003 0196-0709/Published by Elsevier Inc.
AM ER IC AN JOURNAL OF OT OLARYNGOLOGY–H E A D A N D NE CK M E D IC IN E A ND S U RGE RY 3 7 (2 0 1 6) 1 28–1 3 1
caloric expenditure due to increased work of breathing, and alterations of the normal nocturnal growth hormone secretion can contribute to poor weight gain [2–6]. Laryngomalacia is the most common congenital pediatric airway abnormality as well as the most common cause of stridor in infants [7]. Other congenital laryngeal obstructions are bilateral vocal cord paralysis, subglottic stenosis, and cysts [8]. Anatomical manifestations of laryngomalacia typically entail an omega shaped epiglottis, shortened aryepiglottic folds, and redundant arytenoid mucosa and are commonly treated conservatively with a combination of observation, anti-reflux therapy, proper positioning during feeds, prolonged burping, and prone positioning in bed [9–11]. The indications for diagnostic airway evaluation with the potential for surgical intervention vary but are typically considered in cases of severe stridor, apparent life-threatening events (ALTEs), respiratory insufficiency, feeding difficulties, and failure to thrive [12,13]. Techniques for surgical correction of laryngomalacia, known as supraglottoplasty, include but are not limited to division of the aryepiglottic folds, reduction of redundant supraglottic tissue and arytenoid mucosa, and epiglottopexy. This in effect increases airway diameter thus improving stridor and decreasing work of breathing. The procedure is commonly performed with sharp instrumentation such as the laryngeal microscissors, a laser, or a combination of both [12–17]. Many studies have documented weight gain in children after adenotonsillectomy to improve obstructive sleep apnea, but few have evaluated the effects of relief of other forms of airway obstruction on growth patterns. Our objective was to evaluate the short and long term weight changes in children undergoing supraglottoplasty for laryngomalacia to determine if monitoring weight for age percentile growth curves is a sensitive objective parameter for evaluating operative outcomes after supraglottoplasty. All children who had symptomatic laryngomalacia, clinically judged to be severe enough to warrant surgical intervention were treated surgically. Children with a second airway compromise in addition to laryngomalacia, regardless of its severity, were excluded. In clinical practice, pediatric ENT subspecialists are trained to detect and judge the severity of obstruction caused by abnormal laryngeal dynamics during an airway examination. Once the surgeon is convinced of the severity of the pathology, surgical treatment (supraglottoplasty) becomes the choice of treatment. It is the careful selection of the severe subset of pathology by the experienced subspecialist that provides good results in surgical practice. Withholding treatment, therefore allowing for a matched control group is not feasible or ethical for treatments that are known to work, and work well. Age matched children without airway problems were used (standardized growth curves of American children) as an objective measure of catch up growth following surgery.
2.
Methods
An institutional review board approved (Louisiana State University Shreveport STUDY00000270) retrospective chart review of patients who underwent a supraglottoplasty by the otolaryngology department at a single institution from 2/28/ 2012 to 10/20/2014 was performed. Patients were identified
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using the search function of an electronic medical record for procedure CPT codes corresponding to supraglottoplasty (31780 excision tracheal stenosis or 31999 larynx unlisted procedure). Operative notes were reviewed and patients who underwent a traditional supraglottoplasty with scissors and/ or laser for laryngomalacia were included. Patients were excluded if any additional airway procedure was performed at the time of surgery or within the 3 year period postop, such as an adenoidectomy, tonsillectomy, tracheostomy, etc. Patients were also excluded if they had severe co-morbidities or co-morbidities with failure to thrive, such as diagnosed syndromes, chromosomal abnormalities, or neurological deficits. The patient population is defined through limiting clinical variables to enable measurement of treatment effects. Thirty seven charts were identified and reviewed. Four patients who did not have a supraglottoplasty were excluded. Twelve were excluded due to significant medical co-morbidities and/or other airway surgeries at the time of procedure or during the postoperative period. One patient was excluded as no weight data were available. Twenty patients met inclusion and exclusion criteria and were included in the study. Ages of patients meeting criteria ranged from 18 days to 3 years old. 15 (75%) were male and 5 (25%) were female. Nine (45%) were African American, 8 (40%) were Caucasian, and 3 (15%) were of other races. The average weight at preop on CDC sex adjusted growth curves for weight by age was 29.8 percentile. Six patients (30%) met failure-to-thrive criteria by weight less than 5 percentile. Supraglottoplasty was performed with either laryngeal microscissors and/or laser. Microscissors alone were used in 17 cases, 2 cases utilized a combination of scissors and laser, and one case used the laser alone. Weights were recorded for preoperative, 2 week to 3 month postop, 3 month to 1 year postop, and 1 year to 3 year postop intervals. The last recorded weight within each interval was used. 18 patients had weight data in within the first 3 months, 9 patients within 1 year, and 7 within 1–3 years. Paired t-test was used to compare the changes in weight chart percentiles compared to preop at each time interval and p-values and confidence intervals were calculated. The standard p-value of .05 was considered statistically significant.
3.
Results
For many patients weight percentile gain was noted after supraglottoplasty (Fig. 1). At 3 months postop average weight had increased by 7.67 percentile (p = 0.09, 95% CI − 1.62 to 17.0). At 12 months postop average weight had increased by 19.1 percentile (p = 0.06, 95% CI 0.47–37.8). At 3 years postop average weight had increased by 26.53 percentile (p = 0.03, 95% CI 4.47–48.59). By three years postop the average weight had normalized (64.5 percentile). Interestingly among those who met preoperative failure to thrive criteria (average 0.11 percentile), weight gain was still dramatic with average weight percentile gain of 37.5 percentile by 3 years postop.
4.
Discussion
Surgical relief of airway obstruction has previously been reported to be associated with weight gain in the pediatric
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AM ER IC AN JOURNAL OF OT OLA RYNGOLOGY–H E A D A N D NE CK M E D ICI N E AN D S U RGE RY 3 7 (2 0 1 6) 1 28 –1 3 1
Fig. 1 – Weight percentile changes after supraglottoplasty.
patient population. The majority of studies to date have focused on adenotonsillectomy [3–6]. Few studies have evaluated possible weight changes after surgical intervention for laryngomalacia [16,17]. Whymark et al. utilized weight percentiles as a benchmark for measuring outcomes after laser supraglottoplasty. A case–control study by Meier et al. revealed that patients undergoing surgery as opposed to observation or medical therapy often had significantly lower weights at the time of diagnosis. Statistically significant weight gain was noted within the first 3 months, noting a “catch up” growth phase that typically occurs in the immediate postop period. Our data reflected this trend. Weight gain did occur within the first 3 months, though this was not significant at the p = 0.05 level. While Whymark's study showed somewhat of a “plateau” in the 3–6 month interval, our data showed that weight gain continues to occur and that weight gain significantly increases by 3 years postop. This shows that in terms of weight gain, supraglottoplasty for improvement of laryngeal obstruction has both immediate and long term effects. Like the Meier et al. study, many of our patients were lost to followup after the initial postop period and we agree with their prediction that many were likely lost due to significant improvement that no longer required surveillance by a pediatric otolaryngologist. There are several limitations of the study. It is a retrospective review and is not randomized or blinded and as a retrospective study there is potential for selection bias. As only patients who underwent the procedure were reviewed, there was no standard control population. The ramifications with potentially withholding lifesaving treatment make a true control population difficult to establish. However the standardized growth chart percentiles of American children in and of themselves serve as a sort of “control” as the subjects' weights are being tracked compared to age matched normal children. Some patients were not included in the study or within the various time intervals because followup weights
were not available. A significant percentage of the pediatric population served by a pediatric otolaryngologist, particularly those with airway abnormalities such as laryngomalacia, often have severe concomitant medical co-morbidities. As these patients were excluded from the study, the results cannot be generalized for those with severe medical co-morbidities. In our practice, patients who had a history of ALTE, cyanotic events, sleep apnea, feeding difficulties, and FTT as well as patients who displayed signs of respiratory distress during clinical evaluation are scheduled to have an airway evaluation under general anesthesia. While some practitioners like to identify distinct domains of airway obstructions and feeding difficulties in babies and may recommend separate evaluations for each domain, our philosophy does not isolate the effects of airway obstruction on feeding functions. When an airway compromise is present, feeding difficulties ensue. It is rarely appropriate to try to increase the weight before addressing obvious airway compromise, if it is feasible to do so. Therefore, even if the FTT is caused by feeding difficulties, the inciting event is likely to be airway compromise. In this study patients who underwent supraglottoplasty for laryngomalacia were typically significantly underweight for age. Statistically significant weight gain occurred in children after undergoing supraglottoplasty. Future prospective studies should be undertaken to assess when this growth phase is most pronounced and compare results to nonsurgical management if possible. In a typically small-for-age population, surgical intervention for laryngomalacia can normalize growth chart trajectories by 3 years postoperatively, even in children with failure to thrive. FTT is defined as 5th percentile, however almost all patients had lower weight percentiles with an average of 29.8%. This has increased 37.5 points to reach age matched normalized percentiles after surgery. Therefore it is important for the pediatrician, or any
AM ER IC AN JOURNAL OF OT OLARYNGOLOGY–H E A D A N D NE CK M E D IC IN E A ND S U RGE RY 3 7 (2 0 1 6) 1 28–1 3 1
medical provider that works with children, to keep in mind anatomical airway obstructions as a reversible contributor to failure-to-thrive and poor growth chart performance and consider otolaryngology evaluation where indicated. In our tertiary referral pediatric ENT practice, postoperative follow-up and monitoring of children after supraglottoplasty are routinely performed 2–4 weeks and 3 months postoperatively. Caregivers are instructed to report to our clinic whenever they observe respiratory or feeding difficulties, ALTEs, cyanosis, and poor weight gain. Monitoring of overall development and health by their PCP during the first year is also emphasized. The success of the operative treatment is assessed by the parameters above that are subjective and prone to reporting bias by the caregivers with the exception of weight gain. We find that monitoring weight gain and growth curves (percentiles) for weight and for age to be a sensitive clinical indicator of improvement. REFERENCES
[1] Cole SZ, Lanham JS. Failure to thrive: an update. Am Fam Physician 2011;87:829–34. [2] Greenfeld M, Tauman R, DeRowe A, et al. Obstructive sleep apnea syndrome due to adenotonsillar hypertrophy in infants. Int J Pediatr Otorhinolaryngol 2003;67:1055–60. [3] Katz ES, Moore RH, Rosen CL, et al. Growth after adenotonsillectomy for obstructive sleep apnea: an RCT. Pediatrics 2014;134:282–9. [4] Selimoglu E, Selimoglu MA, Orbak Z. Does adenotonsillectomy improve growth in children with obstructive adenotonsillar hypertrophy? J Int Med Res 2003;31:84–7 [4].
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[5] Williams EF, Woo P, MIller R, et al. The effects of adenotonsillectomy on growth in children. Otolaryngol Head Neck Surg 1991;104:509–16 [5]. [6] Yilmaz MD, Hosal AS, Oguz H, et al. The effects of tonsillectomy and adenoidectomy on serum IGF-I and IGFBP3 levels in children. Laryngoscope 2002;112:922–5 [16]. [7] Zoumalan R, Maddalozzo J, Holinger LD. Etiology of stridor in infants. Ann Otol Rhinol Laryngol 2007;116:329–34. [8] Daniel SJ. The upper airway: congenital malformations. Paediatr Respir Rev 2006;7:S260–3. [9] Thompson DM. Abnormal sensorimotor integrative functions of the larynx in congenital laryngomalacia: a new theory of etiology. Laryngoscope 2007;117:1–33. [10] Giannoni C, Sulek M, Friedman E. Gastroesophageal reflux associatd with laryngomalacia: a prospective study. Int J Pediatr Otorhinolaryngol 1998;43:11–20. [11] Olney DR, Greingwald Jr JH, Smith RH, et al. Laryngomalacia and its treatment. Laryngoscope 1999;109:1770–5. [12] Roger, et al. Severe laryngomalacia: surgical indications and results in 115 patients. Laryngoscope 1995;105: 1111–7. [13] Richter GT, Thompson DM. The surgical management of laryngomalacia. Otolaryngol Clin N Am 2008;41:837–64. [14] Lee KS, Chen BN, Yang CC, et al. CO2 laser supraglottoplasty for severe laryngomalacia: a study of symptomatic improvement. Int J Pediatr Otorhinolaryngol 2007;71:889–95. [15] Loke D, Ghosh S, Panarese A, et al. Endoscopic division of the ary-epiglottic folds in severe laryngomalacia. Int J Pediatr Otorhinolaryngol 2001;60:59–63. [16] Whymark AD, Clement WA, Kubba H, et al. Laser epiglottopexy for laryngomalacia: 10 years' experience in the West of Scotland. Arch Otolaryngol Head Neck Surg 2006;132:978–82. [17] Meier JD, Nguyen SA, White DR. Improved growth curve measurements after supraglottoplasty. Laryngoscope 2011; 121:1574–7.