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

Background: Numerous studies have proven the efficacy of mandibular distraction osteogenesis (MDO) or tongue-lip adhesion (TLA) in Robin sequence (RS) infants with upper airway obstruction. However, none have compared health-related quality of life (HRQoL) outcomes post-MDO to post-TLA. Methods: In the present retrospective study, RS-infants younger than 1-year-old, who underwent MDO or TLA, were included (2006–2016). The infants' caregivers were asked to complete a questionnaire based on the Glasgow Children's Benefit Inventory (GCBI). Results: The response rate was 71% (22 of the 31 questionnaires, MDO 12/15 and TLA 10/16), median follow-up was 5.9 years (range:1.3–10.5 years), and median age at surgery was 24 days (range: 5–131 days). The median total GCBI-scores post-MDO and post-TLA were 21.9 (9.4 IQR) and 26.0 (37.5 IQR) respectively (p = 0.716), indicating an overall benefit in HRQoL from both surgical procedures. Positive changes were observed in all subgroups (emotion, physical health, learning, and vitality) for both MDO and TLA. In syndromic RS, both surgical procedures demonstrated a lower positive change in HRQoL compared to isolated RS (19.8 MDO and 16.7 TLA versus 24.0 MDO and 35.4 TLA, p = 0.303). Conclusions: Both MDO and TLA, demonstrated an overall benefit in HRQoL-outcomes, with no significant differences between these two surgical techniques. Our findings contribute to the debate regarding the use of MDO versus TLA in the surgical treatment of RS; however, long-term outcome studies evaluating HRQoL in larger RS-cohorts are necessary to identify which procedure is likely to be best in each individual RS-infant. Disclosure: None of the authors report any conflicts of interest. This research did not receive any funding. None of the authors has a financial interest in any of the products, devices, or drugs mentioned in this manuscript. Medical ethical board approval: This study was approved by the medical ethical board of the University Medical Center Utrecht, The Netherlands (16/647). Acknowledgements: The authors would like to acknowledge Henk F. Van Stel who served as biostatistician for this study. Corresponding Author: Robrecht J.H. Logjes MD, PhD candidate, Department of Plastic and Reconstructive Surgery, University Medical Centre Utrecht, Wilhelmina Children's Hospital, 3508 AB Utrecht, The Netherlands. Email: r.j.h.logjes@gmail.com, Tel. +31 88 77 545 72 ©2019American Society of Plastic Surgeons

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