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Severe obstructive sleep apnea in children with syndromic craniosynostosis: analysis of pulse transit time
Sumin Yang1, Eris van Twist2, Gwen G M van Heesch2
1Department of Plastic and Reconstructive Surgery and Hand Surgery, Erasmus MC Sophia Children's Hospital, Erasmus Medical Center, Rotterdam, The Netherlands.
Insights
Pulse transit time (PTT) shows transient reductions during obstructive events in children with syndromic craniosynostosis (SCS). However, PTT has limited sensitivity and specificity for detecting obstructive sleep apnea (OSA) in this population.
Area of Science:
- Pediatric Cardiology
- Sleep Medicine
- Genetics and Rare Diseases
Background:
- Obstructive sleep apnea (OSA) is a common complication in children with syndromic craniosynostosis (SCS).
- OSA can lead to significant cardiorespiratory disturbances.
- Pulse transit time (PTT) is a potential non-invasive marker for cardiorespiratory changes.
Purpose of the Study:
- To investigate the association between PTT and OSA in pediatric patients with SCS.
- To evaluate the diagnostic utility of PTT for detecting OSA in this specific cohort.
Main Methods:
- Retrospective analysis of 68 children (<18 years) with SCS, categorized by moderate-to-severe OSA or no OSA, and controls.
- Overnight polysomnography was used to diagnose OSA and assess sleep stages.
- Nonparametric bootstrap analysis computed PTT reference intervals; sensitivity and specificity were determined.
Main Results:
- Obstructive events occurred across all sleep stages, with PTT reductions observed 4-8 seconds post-event (P < .05).
- Children with SCS and OSA exhibited lower PTT intervals compared to those without OSA.
- Highest sensitivity for PTT in OSA detection was 55.5% during N1 sleep; highest specificity was 76.5% during REM sleep.
Conclusions:
- Transient PTT reductions accompany obstructive events throughout sleep in children with SCS.
- PTT demonstrates limited sensitivity and specificity, restricting its use as a standalone diagnostic tool for OSA in this population.
Study Objectives:
We examined the association between pulse transit time (PTT) and obstructive sleep apnea (OSA) in children with syndromic craniosynostosis (SCS), where OSA is a common problem and may cause cardiorespiratory disturbance.
Methods:
A retrospective study of children (age < 18 years) with SCS and moderate-to-severe OSA (ie, obstructive apnea-hypopnea index ≥ 5) or no OSA (obstructive apnea-hypopnea index < 1) who underwent overnight polysomnography. Children without SCS and normal polysomnography were included as controls. Reference intervals for PTT were computed by nonparametric bootstrap analysis. Based on reference intervals of controls, the sensitivity and specificity of PTT to detect OSA were determined. In a linear mixed model, the explanatory variables assessed were sex, age, sleep stage, and time after obstructive events.
Results:
In all 68 included children (19 with SCS with OSA, 30 with SCS without OSA, 19 controls), obstructive events occurred throughout all sleep stages, most prominently during rapid eye movement (REM) sleep and non-REM sleep stages N1 and N2, with evident PTT changes. The greatest reductions were observed 4-8 seconds after an event (P < .05). In SCS with OSA, PTT reference intervals were lower during all sleep stages compared with SCS without OSA. The highest sensitivity was observed during N1 (55.5%), and the highest specificity during REM sleep (76.5%). The lowest PTT values were identified during N1.
Conclusions:
Obstructive events occur throughout all sleep stages with transient reductions in PTT. However, PTT as a variable for OSA detection is limited by its sensitivity and specificity.
Citation:
Yang S, van Twist E, van Heesch GGM, et al. Severe obstructive sleep apnea in children with syndromic craniosynostosis: analysis of pulse transit time. J Clin Sleep Med. 2024;20(8):1233-1240.
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