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Drug-induced sleep endoscopy in children with Prader-Willi syndrome

  • ENT • Original Article
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Abstract

Purpose

Review drug-induced sleep endoscopy (DISE) findings in children with Prader-Willi syndrome (PWS) and correlate the patterns of airway collapse with apnea-hypopnea index (AHI) and body mass index (BMI).

Methods

A total of nine children with PWS underwent DISE. DISE findings were recorded using the VOTE classification system. The relationship between different patterns of airway collapse with AHI and BMI was analyzed.

Results

The majority of children with PWS were found to have multilevel obstruction (six out of nine children, 66.6 %). The velum was the most common site of obstruction (nine out of nine children, 100 %). All of the patients had positional obstructive sleep apnea (OSA). Patients with partial or complete anterior-posterior tongue base collapse were associated with a significantly higher AHI (P = 0.016) compared to patients with no anterior-posterior tongue base collapse. Apart from tongue base collapse, no other patterns of airway collapse showed a consistent association with AHI in our results. No patterns of airway collapse showed a significant association with BMI in our study.

Conclusions

In our study, partial or complete anterior-posterior tongue base collapse was associated with higher AHI values in children with PWS. Therefore, careful attention should be addressed to the management of tongue base collapse. Positional therapy could be a potential treatment for patients with PWS since it may alleviate the severity of tongue base collapse.

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Authors and Affiliations

  1. Department of Otolaryngology-Head and Neck Surgery, Taipei Tzu Chi Hospital, Buddhist Tzu Chi Medical Foundation, New Taipei City, Taiwan

    Ming-Chin Lan, Tsan-Jen Chiu, Tung-Tsun Huang & Yu-Cheng Chen

  2. School of Medicine, Tzu Chi University, Hualien, Taiwan

    Ming-Chin Lan, Tsan-Jen Chiu, Tung-Tsun Huang, Shi-Bing Wong, Yu-Cheng Chen & Li-Ping Tsai

  3. Department of Otolaryngology-Head and Neck Surgery, Taipei Veterans General Hospital, Taipei, Taiwan

    Yen-Bin Hsu & Ming-Ying Lan

  4. School of Medicine, National Yang-Ming University, Taipei, Taiwan

    Yen-Bin Hsu & Ming-Ying Lan

  5. Department of Pediatrics, Taipei Tzu Chi Hospital, Buddhist Tzu Chi Medical Foundation, No. 289, Jianguo Rd., Xindian Dist., New Taipei City, 231, Taiwan

    Shi-Bing Wong & Li-Ping Tsai

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  1. Ming-Chin Lan
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  2. Yen-Bin Hsu
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Correspondence to Li-Ping Tsai.

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Ethical approval

All procedures performed in studies involving human participants were in accordance with the ethical standards of the institutional and/or national research committee and with the 1964 Helsinki Declaration and its later amendments or comparable ethical standards.

Conflict of interest

All authors certify that they have no affiliations with or involvement in any organization or entity with any financial interest (such as honoraria; educational grants; participation in speakers’ bureaus; membership, employment, consultancies, stock ownership, or other equity interest; and expert testimony or patent-licensing arrangements) or nonfinancial interest (such as personal or professional relationships, affiliations, knowledge, or beliefs) in the subject matter or materials discussed in this manuscript.

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No funding was received for this research.

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Informed consent was obtained from all individual participants included in the study.

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Lan, MC., Hsu, YB., Lan, MY. et al. Drug-induced sleep endoscopy in children with Prader-Willi syndrome. Sleep Breath 20, 1029–1034 (2016). https://doi.org/10.1007/s11325-016-1338-8

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  • DOI: https://doi.org/10.1007/s11325-016-1338-8

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