Sleep-disordered breathing in children with thoracic insufficiency syndrome

  1. Amanda Striegl MD1,*,
  2. Maida L. Chen MD1,
  3. Yemiserach Kifle MD1,
  4. Kit Song MD2,
  5. Gregory Redding MD1

Article first published online: 2 APR 2010

DOI: 10.1002/ppul.21197

Issue

Pediatric Pulmonology

Pediatric Pulmonology

Volume 45, Issue 5, pages 469–474, May 2010

Additional Information

How to Cite

Striegl, A., Chen, M. L., Kifle, Y., Song, K. and Redding, G. (2010), Sleep-disordered breathing in children with thoracic insufficiency syndrome. Pediatric Pulmonology, 45: 469–474. doi: 10.1002/ppul.21197

Author Information

  1. 1

    Division of Pulmonary and Sleep Medicine, Department of Pediatrics, Seattle Children's Hospital, University of Washington School of Medicine, Seattle, Washington

  2. 2

    Division of Orthopedics, Department of Pediatric Surgery, Seattle Children's Hospital, University of Washington School of Medicine, Seattle, Washington

*Pulmonary Division, Seattle Children's Hospital, MS A-5937, 4800 Sand Point Way NE, Seattle, WA 98105.

Publication History

  1. Issue published online: 21 APR 2010
  2. Article first published online: 2 APR 2010
  3. Manuscript Revised: 20 NOV 2009
  4. Manuscript Accepted: 20 NOV 2009
  5. Manuscript Received: 31 JUL 2009

Funded by

  • Maternal Child Health Bureau. Grant Number: T72MC000007

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Keywords:

  • spine;
  • thorax;
  • scoliosis;
  • congenital;
  • polysomnography;
  • sleep-disordered breathing;
  • obstructive sleep apnea;
  • sleep hypopnea;
  • hypoxia

Abstract

Thoracic insufficiency syndrome (TIS) is a collection of chest and spine malformations that results in progressively restrictive pulmonary mechanics and an inability of the thorax to adequately support lung growth. Many children with TIS are too young to perform standard pulmonary function tests, yet need functional assessments of their restrictive thoracic disease. We report on the sleep architecture and frequency of sleep-related breathing abnormalities in 11 children with TIS who underwent overnight polysomnography from retrospective chart review. Ten of 11 (92%) had sleep disordered breathing as defined by currently accepted criteria of apnea–hypopnea index (AHI) >2 events/hr. The median AHI was 4.3 events/hr, with obstructive hypopneas (median 3.7 events/hr) accounting for 75% of abnormalities. Respiratory events occurred most frequently during REM sleep (median REM-AHI 17.3 events/hr), and were associated with oxyhemoglobin desaturation, and rarely carbon dioxide retention. Sleep disordered breathing with hypoxemia appears to be a common but under recognized problem among children with TIS. Polysomnogram may have a role as a non-invasive screening tool used in conjunction with other functional respiratory assessments in children with TIS, and warrants further study in a prospective manner. Pediatr Pulmonol. 2010; 45:469–474. © 2010 Wiley-Liss, Inc.

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