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Mid-Term Dental and Nerve-Related Complications of Infant Distraction for Robin Sequence

van Nunen, Daan P. F. M.D.; Breugem, Corstiaan C. M.D., Ph.D.

Plastic and Reconstructive Surgery: March 2017 - Volume 139 - Issue 3 - p 805e–806e
doi: 10.1097/PRS.0000000000003099

Division of Pediatric Plastic Surgery, Wilhelmina Children’s Hospital, University Medical Center Utrecht, Utrecht, The Netherlands

Correspondence to Dr. van Nunen, Division of Pediatric Plastic Surgery, Wilhelmina Children’s Hospital, University Medical Center Utrecht, Heidelberglaan 100, P.O. Box 85500, 3508 GA Utrecht, The Netherlands,

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We would like to congratulate Steinberg et al. for their article “Mid-Term Dental and Nerve-Related Complications of Infant Distraction for Robin Sequence” published in Plastic and Reconstructive Surgery in July of 2016.1 The authors demonstrated a significant incidence of mid-term dental and nerve-related complications following internal mandibular distraction for airway obstruction in patients with Robin sequence. To distinguish iatrogenic dental injuries from possible congenital dental anomalies, the authors listed the presence of “craniofacial dysmorphic syndromes” as an exclusion criterion in the Patients and Methods section. However, the results show that 14 patients (or 16.5 percent of the total) had additional congenital anomalies in the form of either Stickler syndrome, chromosomal defects, or other organ system/limb malformations. The article does not explain why these patients were still included. More importantly, the study provides no information regarding the manner in which the included patients were assessed for possible congenital anomalies. Were all patients routinely examined by a medical geneticist? Furthermore, have the initial diagnoses of the medical geneticist(s) been reevaluated in later years?

We feel such information is relevant for the correct interpretation of the study results for two reasons. First, a recent thorough reexamination of the medical records of a large cohort of Dutch patients with Robin sequence showed that in only 37.7 percent of patients could the condition be considered as truly isolated and thus without a concomitant syndrome, chromosomal anomaly, or spectrum of malformations.2 Moreover, reevaluation by a panel of medical geneticists changed the initial diagnosis in a quarter of the patients examined, reducing the number classified as “isolated” Robin sequence. Second, anomalies of the dentition are associated with a host of recognized syndromes.3 The two overviews of tooth agenesis in isolated Robin sequence referred to in the article (Antonarakis and Suri4 and Andersson et al.5) therefore specifically exclude all patients with craniofacial malformations or any additional syndromes (including Stickler syndrome). We hope that Plastic and Reconstructive Surgery will allow the authors the opportunity to respond to improve our understanding of their important study.

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The authors have no financial interest to declare in relation to the content of this communication.

Daan P. F. van Nunen, M.D.

Corstiaan C. Breugem, M.D., Ph.D.

Division of Pediatric Plastic Surgery

Wilhelmina Children’s Hospital

University Medical Center Utrecht

Utrecht, The Netherlands

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1. Steinberg JP, Brady CM, Waters BR, et alMid-term dental and nerve-related complications of infant distraction for Robin sequence.Plast Reconstr Surg201613882e–90e
2. Basart H, Paes EC, Maas SM, et alEtiology and pathogenesis of Robin sequence in a large Dutch cohort.Am J Med Genet A20151671983–1992
3. Klein OD, Oberoi S, Huysseune A, Hovorakova M, Peterka M, Peterkova RDevelopmental disorders of the dentition: An update.Am J Med Genet C Semin Med Genet2013163318–332
4. Antonarakis GS, Suri SPrevalence and patterns of permanent tooth agenesis in patients with nonsyndromic Pierre Robin sequence.Am J Orthod Dentofacial Orthop2014145452–460
5. Andersson EM, Feragen KB, Mikalsen D, Kaul J, Holla TM, Filip CBilateral hypodontia in adolescents with Pierre Robin sequence.Cleft Palate Craniofac J201552452–457
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