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.
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