Infant mandibular distraction in absence of ascending ramus: case series

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CASE REPORT

Infant mandibular distraction in absence of ascending ramus: case series Bruno Brevi 1 & Michela Bergonzani 2

&

Francesca Zito 2 & Andrea Varazzani 2 & Enrico Sesenna 2

Received: 5 September 2019 / Accepted: 10 March 2020 # Springer-Verlag GmbH Germany, part of Springer Nature 2020

Abstract Background Severe microretrognathia with the absence of ascending mandibular ramus is a challenging deformity and treatment must aim to avoid tracheostomy or remove it as soon as possible. Although it is not often reported, mandibular distraction osteogenesis represents a valid treatment option in infants affected by hypoplastic mandible Pruzansky-Kaban type IIb and III. Case presentation The authors describe 3 cases of infants affected by severe respiratory insufficiency due to congenital mandibular hypoplasia, with follow up ranging from 4 to 8 years. Clinical and technical considerations on treatment choices and outcomes are discussed starting from review of the literature and direct clinical experience. Conclusion Early mandibular distraction, specifically bidirectional distraction, is an effective and repeatable technique that leads to mandible lengthening with counterclockwise rotation, pogonion projection increase, anteropositioning of the tongue base, and expansion of oropharyngeal volume with positive effect on the respiratory problems of the infant. Even in Treacher Collins patients, known to have a low decannulation rate, all of these elements are essential for effective speech and swallowing therapy and for a subsequent attempt of decannulation. Keywords Mandibular distraction . Mandibular hypoplasia . Upper airway obstruction . Syndromic anomalies

Introduction Severe congenital microretrognathia may lead to upper airway obstruction in infants and early tracheostomy may be necessary in the most severe cases. Based on the grade of upper airway obstruction and on the type of mandibular hypoplasia, different therapeutic options can be considered. Mandibular hypoplasia with the absence of ascending ramus, often associated with the absence of glenoid fossa, is a complex and challenging deformity to approach. Treatment must aim to avoid tracheostomy or remove it as soon as possible. Some of the surgical treatments for microretrognathia are not feasible when dealing with infants with absent mandibular ramus. Due to the mandibular anomaly itself, classic Obwegeser

* Michela Bergonzani [email protected] 1

Complex Operating Unit of Maxillo-Facial Surgery, Azienda Ospedaliero-Universitaria Pisana, Pisa, Italy

2

Maxillo-Facial Surgery Division, Head and Neck Department, University Hospital of Parma, via Gramsci 14, 43100 Parma, Italy

sagittal split and inverted L osteotomy to obtain elongation of the mandibular body or of the ramus cannot be performed. According to the literature, severe micrognathia can be managed with costochondral grafting, distraction osteogenesis, vascularized tissue transfer, titanium prosthesis, or combination of techniques. Despite the fact that different authors do not