Maxillary hypoplasia is a major issue in cleft lip and palate patients, and predictable surgical maxillary advancement is required. In the present study, the changes and stability of the maxilla and soft tissue profile achieved after the application of anterior maxillary distraction osteogenesis (AMDO) using intraoral expander in unilateral cleft lip and palate and isolated cleft palate patients were investigated by comparing to the Le Fort I osteotomy (LFI) and maxillary distraction osteogenesis (DO) with rigid external distraction (RED) system.Ten patients who underwent orthognathic treatment with AMDO were examined (AMDO group). Changes in the positions of soft and hard tissue landmarks were calculated from the lateral cephalograms taken before the distraction, at the end of the distraction, and 1 year after the surgery. They were compared with the changes in 7 other unilateral cleft lip and palate patients who underwent LFI (LFI group) and 6 others who underwent DO with RED (RED group).The mean maxillary advancement of the AMDO group was similar to that of the RED group, judged by the change of point A. During DO, the AMDO group showed less clockwise rotation of mandible compared to the RED group. The soft tissue advancement of the upper lip and nose in the AMDO group was similar to that in the RED group, which was significantly larger than that in the LFI group.Our results indicate that AMDO can be surgical option to cleft lip and palate patients with less invasive but excellent improvement in both midfacial skeletal and soft tissue similar to DO-RED.
Anterior maxillary distraction osteogenesis (AMDO) is a novel technique for correcting hypoplastic maxilla by sagittal expansion of the maxilla. Recent reports suggest that AMDO does not have an effect on fragile velopharyngeal function in patients with cleft palate. Furthermore, no studies have evaluated the impact of AMDO on velopharyngeal function. We adopted AMDO to correct severe hypoplastic maxilla in adolescent patients with cleft palate and evaluated its impact on velopharyngeal space and function in 8 patients aged 12 to 21 years who underwent AMDO from 2006 to 2014. All the patients had received treatment for cleft palate; however, they still exhibited marginal velopharyngeal insufficiency. The mean activation of the distractor was 10.9 ± 0.9 mm. We determined changes in velopharyngeal closure ratio and closure pattern via nasopharyngoscopy. Additionally, skeletal changes were evaluated using lateral cephalograms. The mean horizontal advancement in the cephalogram obtained 1 year after the distraction was +6.4 mm. Nasopharyngoscopic examination revealed that no deterioration of velopharyngeal gap had occurred after AMDO in all 8 patients. The velopharyngeal closure pattern changed from coronal to circular in 1 patient. Our results indicate that AMDO achieved correction of hypoplastic maxilla without deterioration in velopharyngeal gap and function. Therefore, AMDO is an effective and optimal approach for correcting hypoplastic maxilla especially in patients with fragile velopharyngeal function, such as those with cleft palate.
Summary: Hypoglossia-hypodactyly, or aglossia-adactylia syndrome with or without limb anomalies, is an extremely rare congenital condition. It is characterized by a narrow, V-shaped mandibular dental arch with micrognathia, and is typically challenging to treat. We have previously reported 3 patients with hypoglossia-hypodactyly syndrome without limb anomalies who were treated with transverse distraction osteogenesis at the mandibular symphysis during childhood. In this report, we present the long-term prognoses of these 3 cases, until 18 years of age. Of the 3 total cases, sufficient and stable results in occlusion, speech, mastication, and facial appearance were obtained in 2 cases with subsequent orthodontic treatments. Similar results were not observed in the remaining case when continuous orthodontic treatments were not performed. Based on these results, we propose that the transverse distraction osteogenesis procedure at the mandibular symphysis during childhood could be a reliable method to correct the V-shaped mandibular arch of hypoglossia-hypodactyly syndrome, when subsequent orthodontic treatments are provided during the patient’s growing period.
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