2017 Sec 1 Green Book

Reprinted by permission of Laryngoscope. 2017; 127(2):520-524.

The Laryngoscope V C 2016 The American Laryngological, Rhinological and Otological Society, Inc.

Polysomnographic Outcomes Following Lingual Tonsillectomy for Persistent Obstructive Sleep Apnea in Down Syndrome

J. Drew Prosser, MD; Sally R. Shott, MD; Oscar Rodriguez, MD; Narong Simakajornboon, MD; Jareen Meinzen-Derr, PhD; Stacey L. Ishman, MD, MPH

Objectives/Hypothesis: Lingual tonsil hypertrophy is a common cause of persistent airway obstruction in patients with Down syndrome (DS) following adenotonsillectomy (T&A); however, little is known about the effect of lingual tonsillectomy (LT) on polysomnographic outcomes in these patients. Our objective was to describe changes in sleep-related respiratory out- comes following LT in children with DS and persistent obstructive sleep apnea (OSA) following T&A. Study Design: Retrospective case series. Methods: We included all children with DS who underwent polysomnography before and after LT at a tertiary care cen- ter from 2003 to 2013. Nonparametric analysis of variables was performed. Results: Forty patients with DS underwent LT; 21 met inclusion criteria. The mean age at surgery was 9.3 6 4.3 years and 47.6% were female. The median apnea-hypopnea index (AHI) was 9.1 events/hour (range, 3.8 to 43.8 events/hour) before surgery and 3.7 events/hour (range, 0.5 to 24.4 events/hour) after surgery. The median improvement in overall AHI and the obstructive AHI (oAHI) were 5.1 events/hour (range, 2 2.9 to 41) and 5.3 events/hour (range, 2 2.9 to 41), respec- tively ( P < .0001). The mean oxygen saturation nadir improved from 84% to 89% ( P 5 .004). The mean time with CO 2 > 50 mm Hg, central index, and percentage of rapid eye movement sleep were not significantly different. After surgery, the oAHI was < 5 events/hour in 61.9% and 1 in 19% of patients. Conclusions: In children with DS, persistent OSA after T&A and lingual tonsil hypertrophy, LT significantly improved AHI, oAHI, and O 2 saturation nadir. We recommend that children with DS should be evaluated for lingual tonsil hypertrophy if found to have persistent OSA following T&A. Key Words: Obstructive sleep apnea, Down syndrome, lingual tonsil hypertrophy, lingual tonsillectomy. Level of Evidence: 4 Laryngoscope , 00:000–000, 2016

INTRODUCTION Obstructive sleep apnea (OSA) is a sleep-related breathing disorder associated with significant morbidity in children, including impaired memory, behavioral dis- turbances, attention deficit and hyperactivity disorder, and cardiovascular disease. 1,2 Althoughe the prevalence of OSA in the general pediatric population is low Additional Supporting Information may be found in the online version of this article. From the Department of Otolaryngology–Head and Neck Surgery ( J . D . P .), Georgia Regents University, Augusta, Georgia; Division of Pediat- ric Otolaryngology–Head and Neck Surgery ( S . R . S ., S . L . I .), Cincinnati Children’s Hospital Medical Center, Cincinnati, Ohio; Department of Otolaryngology–Head and Neck Surgery ( S . R . S ., S . L . I .), University of Cin- cinnati College of Medicine, Cincinnati, Ohio; Department of Pediatric Pulmonary and Sleep Medicine ( O . R .), University of Mississippi Medical Center, Jackson, Mississippi; Division of Pulmonary and Sleep Medicine ( N . S ., S . L . I .), Cincinnati Children’s Hospital Medical Center, Cincinnati, Ohio; Division of Biostatistics and Epidemiology ( J . M .- D .), Cincinnati Children’s Hospital Medical Center, Cincinnati, Ohio, U.S.A. Editor’s Note: This Manuscript was accepted for publication June 28, 2016. Presented at the Triological Society Combined Sections Meeting, Miami, Florida, U.S.A., January 22–24, 2016. The authors have no funding, financial relationships, or conflicts of interest to disclose. Send correspondence to Stacey L. Ishman, MD, Cincinnati Child- ren’s Hospital Medical Center, 3333 Burnet Avenue, MLC 2018, Cincin- nati, Ohio 45229. E-mail: stacey.ishman@cchmc.org

(approximately 1%–2%), the prevalence is much higher in children with Down syndrome (DS), where an esti- mated 40% to 80% are affected. 3,4 Adenotonsillectomy (T&A) is the first-line treatment in children with OSA; however, persistent OSA is common after surgery in children with DS. Studies have shown that in children with DS, T&A results in resolution of OSA, with an obstructive AHI 1 event/hour, in only 16% to 33% of those treated. 5,6 Several factors have been implicated in the patho- genesis of persistent OSA in patients with DS. These include muscular hypotonia as well as anatomic features such as macroglossia, relative glossoptosis, midface hypoplasia, hypopharyngeal collapse, and lingual tonsil hypertrophy. 7 Anatomic studies have shown lingual ton- sil hypertrophy to be significantly more common in chil- dren with DS than in controls, with almost 35% of children with persistent OSA and DS reported to have lingual tonsil hypertrophy, compared to 3% of the gener- al pediatric population. 8,9 Lingual tonsillectomy (LT) has been shown to improve OSA in patients with lingual tonsil hypertrophy and persistent OSA after T&A. 10 Despite evidence that lingual tonsil hypertrophy plays a significant role in the persistence or recurrent of pediatric OSA, and the recog- nition that this finding is more prevalent in patients with DS than in children without comorbidities, no

DOI: 10.1002/lary.26202

Prosser et al.: PSG Outcomes of Lingual Tonsillectomy in DS

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