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

Steinberg, Jordan P. M.D., Ph.D.; Thomas, Jack E. D.D.S.; Williams, Joseph K. M.D.

Plastic & Reconstructive Surgery: March 2017 - Volume 139 - Issue 3 - p 806e–807e
doi: 10.1097/PRS.0000000000003100

Center for Craniofacial Disorders, Children’s Healthcare of Atlanta at Scottish Rite, Atlanta, Ga.

Correspondence to Dr. Steinberg, Advocate Children’s Hospital, 2 East Pavilion, Box 82, 1675 Dempster Street, Park Ridge, Ill. 60068,

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We would like to acknowledge the insightful commentary provided by van Nunen and Breugem on our recent article.1 The authors have inquired about our decision to include patients with associated anomalies in the analysis such as those with Stickler syndrome, chromosomal translocations, or other organ system/limb malformations. This is questioned specifically with reference to our statement in the Patients and Methods section that “patients with craniofacial dysmorphic syndromes and others affecting development or function of the facial nerves (unilaterally or bilaterally)” were excluded.

First, we would like to point out that all of our Robin sequence patients were indeed evaluated by a geneticist at the time of the initial evaluation in the neonatal intensive care unit. Clinical examinations were accompanied by microarray and/or specific mutation testing where indicated. In terms of reevaluation in later years, we would emphasize that all patients followed in our multidisciplinary clinic were routinely assessed not only by the plastic surgeon, pediatric dentist, and speech language pathologist, but also by the geneticist and psychologist.

With respect to associated anomalies, we felt it appropriate to include those patients with conditions not obviously associated with the complications on which we were focused in this study: tooth maldevelopment and nerve dysfunction. Therefore, patients with conditions such as craniofacial microsomia and Treacher Collins syndrome were excluded, whereas others with balanced chromosomal translocations or limb anomalies and no facial dysmorphism were not excluded. Facial differences may be appreciated in Stickler syndrome and, as the authors have indicated, other groups have excluded such patients from dental analyses.2,3 However, consistent patterns of anomalous dental development have not been reported for this population in the literature to date.

Although a clearer argument in favor of iatrogenic injury may be made in cases featuring gross tooth deformity and unilateral nerve palsy, we acknowledge that, when it comes to the issue of second premolar agenesis, other etiologic factors are difficult to rule out. In reanalyzing our data, we noted that three of our patients with absent second premolars had nonisolated Robin sequence. One with a chromosomal translocation had bilateral absence of the maxillary second premolars. Another with a chromosomal translocation had bilateral absence of the mandibular second premolars. A third patient with Stickler syndrome had unilateral absence of the left mandibular second premolar. Even with the exclusion of these patients, our findings still indicate a predominance of unilateral second premolar agenesis in the mandible with associated distalization of the remaining premolar. This pattern is in distinct contrast to the bilateral agenesis seen with genetic influences or with Robin sequence itself.2,3 As a consequence, our conclusions regarding the potential for tooth and nerve injury and the associated implications remain unchanged.

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

Jordan P. Steinberg, M.D., Ph.D.

Jack E. Thomas, D.D.S.

Joseph K. Williams, M.D.

Center for Craniofacial Disorders

Children’s Healthcare of Atlanta at Scottish Rite

Atlanta, Ga.

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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. Andersson EM, Feragen KB, Mikalsen D, Kaul J, Holla TM, Filip CBilateral hypodontia in adolescents with Pierre Robin sequence.Cleft Palate Craniofac J201552452–457
3. Antonarakis GS, Suri SPrevalence and patterns of permanent tooth agenesis in patients with nonsyndromic Pierre Robin sequence.Am J Orthod Dentofacial Orthop2014145452–460
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