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Observational Study
. 2016 Dec;138(6):1019e-1029e.
doi: 10.1097/PRS.0000000000002741.

Sleep Architecture Linked to Airway Obstruction and Intracranial Hypertension in Children with Syndromic Craniosynostosis

Bart Spruijt  1   2 , Irene M J Mathijssen  1   2 , Hansje H Bredero-Boelhouwer  1   2 , Perumpillichira J Cherian  1   2 , Linda J A Corel  1   2 , Marie-Lise van Veelen  1   2 , Richard D Hayward  1   2 , Robert C Tasker  1   2 , Koen F M Joosten  1   2
Affiliations
Observational Study

Sleep Architecture Linked to Airway Obstruction and Intracranial Hypertension in Children with Syndromic Craniosynostosis

Bart Spruijt et al. Plast Reconstr Surg. 2016 Dec.

Abstract

Background: Children with syndromic craniosynostosis often have obstructive sleep apnea and intracranial hypertension. The authors aimed to evaluate (1) sleep architecture, and determine whether this is influenced by the presence of obstructive sleep apnea and/or intracranial hypertension; and (2) the effect of treatment on sleep architecture.

Methods: This study included patients with syndromic craniosynostosis treated at a national referral center, undergoing screening for obstructive sleep apnea and intracranial hypertension. Obstructive sleep apnea was identified by polysomnography, and categorized into no, mild, moderate, or severe. Intracranial hypertension was identified by the presence of papilledema on funduscopy, supplemented by optical coherence tomography and/or intracranial pressure monitoring. Regarding sleep architecture, sleep was divided into rapid eye movement or non-rapid eye movement sleep; respiratory effort-related arousals and sleep efficiency were scored.

Results: The authors included 39 patients (median age, 5.9 years): 19 with neither obstructive sleep apnea nor intracranial hypertension, 11 with obstructive sleep apnea (four moderate/severe), six with intracranial hypertension, and three with obstructive sleep apnea and intracranial hypertension. Patients with syndromic craniosynostosis, independent of the presence of mild obstructive sleep apnea and/or intracranial hypertension, have normal sleep architecture compared with age-matched controls. Patients with moderate/severe obstructive sleep apnea have a higher respiratory effort-related arousal index (p < 0.01), lower sleep efficiency (p = 0.01), and less rapid eye movement sleep (p = 0.04). An improvement in sleep architecture was observed following monobloc surgery (n = 5; rapid eye movement sleep, 5.3 percent; p = 0.04).

Conclusions: Children with syndromic craniosynostosis have in principle normal sleep architecture. However, moderate/severe obstructive sleep apnea does lead to disturbed sleep architecture, which fits within a framework of a unifying theory for obstructive sleep apnea, intracranial hypertension, and sleep.

Clinical question/level of evidence: Risk, II.

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References

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    1. Doerga PN, Spruijt B, Mathijssen IM, Wolvius EB, Joosten KF, van der Schroeff MP. Upper airway endoscopy to optimize obstructive sleep apnea treatment in Apert and Crouzon syndromes. J Craniomaxillofac Surg. 2016;44:191196.
    1. Hoeve LJ, Pijpers M, Joosten KF. OSAS in craniofacial syndromes: An unsolved problem. Int J Pediatr Otorhinolaryngol. 2003;67(Suppl 1):S111S113.
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