Volume 131, Issue 4 pp. 898-906

Systematic Review

Outcomes of Adenotonsillectomy for Obstructive Sleep Apnea in Prader-Willi Syndrome: Systematic Review and Meta-analysis

Anna Christina Clements BS,

Anna Christina Clements BS

orcid.org/0000-0002-6869-3339

Johns Hopkins University School of Medicine, Baltimore, Maryland, U.S.A.

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Xi Dai BA,

Xi Dai BA

Johns Hopkins University School of Medicine, Baltimore, Maryland, U.S.A.

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Jonathan M. Walsh MD,

Jonathan M. Walsh MD

Department of Otolaryngology–Head and Neck Surgery, Johns Hopkins University, Baltimore, Maryland, U.S.A.

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Laura M. Sterni MD,

Laura M. Sterni MD

Eudowood Division of Pediatric Respiratory Sciences, Johns Hopkins University School of Medicine, Baltimore, Maryland, U.S.A.

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Laura Prichett PhD, MHS,

Laura Prichett PhD, MHS

Biostatistics, Epidemiology and Data Management (BEAD) Core, Johns Hopkins School of Medicine, Baltimore, Maryland, U.S.A.

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Emily F. Boss MD, MPH,

Emily F. Boss MD, MPH

Department of Otolaryngology–Head and Neck Surgery, Johns Hopkins University, Baltimore, Maryland, U.S.A.

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Stella M. Seal MLS,

Stella M. Seal MLS

Johns Hopkins University School of Medicine, Welch Medical Library, Baltimore, Maryland, U.S.A.

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Marisa A. Ryan MD, MPH,

Corresponding Author

Marisa A. Ryan MD, MPH

[email protected]

Department of Otolaryngology–Head and Neck Surgery, Johns Hopkins University, Baltimore, Maryland, U.S.A.

Send correspondence to Marisa A. Ryan MD, MPH, 601 North Caroline Street, Baltimore, MD 21287. E-mail: [email protected]Search for more papers by this author

Anna Christina Clements BS,

Anna Christina Clements BS

orcid.org/0000-0002-6869-3339

Johns Hopkins University School of Medicine, Baltimore, Maryland, U.S.A.

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Xi Dai BA,

Xi Dai BA

Johns Hopkins University School of Medicine, Baltimore, Maryland, U.S.A.

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Jonathan M. Walsh MD,

Jonathan M. Walsh MD

Department of Otolaryngology–Head and Neck Surgery, Johns Hopkins University, Baltimore, Maryland, U.S.A.

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Laura M. Sterni MD,

Laura M. Sterni MD

Eudowood Division of Pediatric Respiratory Sciences, Johns Hopkins University School of Medicine, Baltimore, Maryland, U.S.A.

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Laura Prichett PhD, MHS,

Laura Prichett PhD, MHS

Biostatistics, Epidemiology and Data Management (BEAD) Core, Johns Hopkins School of Medicine, Baltimore, Maryland, U.S.A.

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Emily F. Boss MD, MPH,

Emily F. Boss MD, MPH

Department of Otolaryngology–Head and Neck Surgery, Johns Hopkins University, Baltimore, Maryland, U.S.A.

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Stella M. Seal MLS,

Stella M. Seal MLS

Johns Hopkins University School of Medicine, Welch Medical Library, Baltimore, Maryland, U.S.A.

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Marisa A. Ryan MD, MPH,

Corresponding Author

Marisa A. Ryan MD, MPH

[email protected]

Department of Otolaryngology–Head and Neck Surgery, Johns Hopkins University, Baltimore, Maryland, U.S.A.

Send correspondence to Marisa A. Ryan MD, MPH, 601 North Caroline Street, Baltimore, MD 21287. E-mail: [email protected]Search for more papers by this author

First published: 27 July 2020

https://doi.org/10.1002/lary.28922

Citations: 17

Editor's Note: This Manuscript was accepted for publication on June 18, 2020.

Disclosures: Dr. Emily Boss is an editorial board member for tonsillectomy decision aids for Option Grid/EBSCO Health.

This research was selected for presentation at the Triological Society Combined Sections Meeting on January 23-25, 2020 in Coronado, CA, USA.

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Abstract

Objectives

Prader-Willi syndrome (PWS) increases the risk of obstructive sleep apnea (OSA) due to obesity, hypotonia, and abnormal ventilatory responses. We evaluated post-adenotonsillectomy complications, polysomnography changes, and quality of life in children with OSA and PWS.

Study Design

Systematic review and meta-analysis.

Methods

We conducted a systematic review and meta-analysis by searching PubMed, Embase, Cochrane, Web of Science, and Scopus. Two researchers independently reviewed studies about adenotonsillectomy for OSA in patients <21 years with PWS. We extracted study design, patient numbers, age, complications, polysomnography, and quality of life. We pooled postoperative changes in apnea hypopnea index (AHI) for meta-analysis. We applied Methodological Index for Nonrandomized Studies (MINORS) criteria to assess study quality.

Results

The initial search yielded 169 studies. We included 68 patients from eight studies with moderate to high risk of bias. Six studies reported on complications and 12 of 51 patients (24%) had at least one. Velopharyngeal insufficiency was the most commonly reported complication (7/51, 14%). We included seven studies in meta-analysis. Mean postoperative improvement in AHI was 7.7 (95% CI: 4.9–10.5). Postoperatively 20% (95% CI: 3%–43%) had resolution of OSA with AHI < 1.5 while 67% (95% CI: 50%–82%) had improvement from severe/moderate OSA to mild/resolved (AHI < 5). Two studies evaluated quality of life and demonstrated improvement.

Conclusions

Children with PWS undergoing adenotonsillectomy for OSA have a substantial risk of postoperative complications that may require additional interventions, especially velopharyngeal insufficiency. Despite improvements in polysomnography and quality of life, many patients had residual OSA. This information can be used to counsel families when considering OSA treatment options. Laryngoscope, 131:898–906, 2021

Supporting Information

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Citing Literature

Volume131, Issue4

April 2021

Pages 898-906

Filename	Description
lary28922-sup-0001-TableS1.docxWord 2007 document , 15 KB	Supporting Table 1 Articles excluded on full-text review and reason for exclusion.
lary28922-sup-0002-Supinfo.docxWord 2007 document , 17.7 KB	Appendix 1. Search strategy conducted in PubMed, Embase, Cochrane, Web of Science, and Scopus using combinations of Medical Subject Headings (MeSH), text terms and keywords.

Outcomes of Adenotonsillectomy for Obstructive Sleep Apnea in Prader-Willi Syndrome: Systematic Review and Meta-analysis

Abstract

Objectives

Study Design

Methods

Results

Conclusions

Supporting Information

BIBLIOGRAPHY

Citing Literature

References

Information

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Outcomes of Adenotonsillectomy for Obstructive Sleep Apnea in Prader-Willi Syndrome: Systematic Review and Meta-analysis

Abstract

Objectives

Study Design

Methods

Results

Conclusions

Supporting Information

BIBLIOGRAPHY

Citing Literature

References

Related

Information