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Assessment of Health-Related Quality of Life in Robin Sequence: A Comparison of Mandibular Distraction Osteogenesis and Tongue-Lip Adhesion

Logjes, Robrecht J. H. M.D.; Mermans, Joline F. M.D.; Paes, Emma C. M.D., Ph.D.; Muradin, Marvick S. M. M.D., D.M.D., Ph.D.; Don Griot, J. Peter M.D., Ph.D.; Breugem, Corstiaan C. M.D., Ph.D.

Plastic and Reconstructive Surgery: May 2019 - Volume 143 - Issue 5 - p 1456-1465
doi: 10.1097/PRS.0000000000005510
Pediatric/Craniofacial: Original Articles
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Background: Numerous studies have proven the efficacy of mandibular distraction osteogenesis or tongue-lip adhesion in Robin sequence infants with upper airway obstruction. However, none has compared health-related quality of life outcomes.

Methods: In the present retrospective study, Robin sequence infants younger than 1 year, who underwent mandibular distraction osteogenesis or tongue-lip adhesion, were included (2006 to 2016). The infants’ caregivers were asked to complete a questionnaire based on the Glasgow Children’s Benefit Inventory.

Results: The response rate was 71 percent (22 of the 31 questionnaires; mandibular distraction osteogenesis, 12 of 15; and tongue-lip adhesion, 10 of 16) and median age at surgery was 24 days (range, 5 to 131 days). Median total Glasgow Children’s Benefit Inventory scores after mandibular distraction osteogenesis and after tongue-lip adhesion were 21.9 (interquartile range, 9.4) and 26.0 (interquartile range, 37.5), respectively (p = 0.716), indicating an overall benefit from both procedures. Positive changes were observed in all subgroups emotion, physical health, learning, and vitality. In syndromic Robin sequence, both procedures demonstrated a lower positive change in health-related quality of life compared with isolated Robin sequence (p = 0.303).

Conclusions: Both surgical procedures demonstrated an overall benefit in health-related quality-of-life outcomes, with no significant differences. The authors’ findings contribute to the debate regarding the use of mandibular distraction osteogenesis versus tongue-lip adhesion in the surgical treatment of Robin sequence; however, studies evaluating health-related quality of life in larger Robin sequence cohorts are necessary to identify which procedure is likely to be best in each individual Robin sequence infant.

CLINICAL QUESTION/LEVEL OF EVIDENCE: Therapeutic, III.

Utrecht, Amsterdam, and Amersfoort, The Netherlands

From the Departments of Plastic and Reconstructive Surgery and Cranio-Maxillofacial Surgery, University Medical Centre Utrecht/Wilhelmina Children’s Hospital; the Department of Plastic and Reconstructive Surgery, Amsterdam University Medical Center, Location VU and AMC; and the Department of Plastic and Reconstructive Surgery, Meander Medical Center.

Received for publication March 22, 2018; accepted August 30, 2018.

Presented at the 32nd Annual Meeting of the Dutch Association of Cleft and Craniofacial Anomalies, in Utrecht, The Netherlands, November 18, 2017; the Second International Robin Sequence Consensus Meeting, in Toronto, Ontario, Canada, May 8, 2017; and the 13th International Cleft Congress, in Chennai, India, February 11, 2017.

Disclosure:The authors have no financial interest to declare in relation to the content of this article. No funding was received.

Robrecht J. H. Logjes, M.D., Department of Plastic and Reconstructive Surgery, University Medical Centre Utrecht, Wilhelmina Children’s Hospital, 3508 AB Utrecht, The Netherlands, r.j.h.logjes-2@umcutrecht.nl

Copyright © 2019 by the American Society of Plastic Surgeons