PubMed · 3907798
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P V Cooney. 1985. Hypodontia. Two case reports.. https://pubmed.ncbi.nlm.nih.gov/3907798/
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X-linked recessive anhidrotic ectodermal dysplasia with immunodeficiency is a developmental and immunologic disorder caused by mutations in nuclear factor-kappaB essential modulator (NEMO), which is essential for nuclear factor-kappaB activation. Early in life, affected boys present a typical appearance, with hypotrichosis or atrichosis, hypohidrosis or anhidrosis, and hypodontia or anodontia with conical incisors. They are also susceptible to various microorganisms, mostly pyogenic bacteria and mycobacteria. Here we report 2 unrelated boys, aged 6 and 11 years, who have novel mutations in NEMO and present conical incisors and hypodontia as their sole and long-unrecognized developmental anomaly. One child had isolated recurrent pneumococcal disease, whereas the other had multiple infections. Our observations indicate that conical incisors should prompt the search for NEMO mutations in boys with unusual infectious diseases.
BACKGROUND: Distraction osteogenesis (DO) has become a mainstream surgical technique for patients with jaw deformities. In cases of maxillary hypoplasia, DO with a rigid external distraction (RED) system has been used for maxillary advancement; however, DO with internal devices is currently popular. MATERIALS: This article describes DO with an internal device and a RED system in 2 patients with maxillary hypoplasia with oligodontia. The first patient, a young girl, had a concave profile due to maxillary hypoplasia and 9 congenitally missing permanent teeth. At age 11 years 11 months, she received DO with an internal device. The second patient, a boy aged 11 years 7 months, was treated with DO with a RED system. RESULTS: In the girl, the maxilla was advanced 5.0 mm without any dentoalveolar compensation. In the boy, the maxilla was advanced 7.0 mm, but undesirable mesial movement of posterior teeth was observed. CONCLUSIONS: DO with internal devices is simpler and more useful than the RED system for maxillary hypoplasia with oligodontia.
When you treat the congenital absence of mandibular second premolars by extracting the residual deciduous second molar and closing the space, you run the risk of flattening facial fullness, especially in patients with nonextraction features--diminished facial fullness, absence of crowding, and reduced tooth size. This challenge is heightened by the increased likelihood that these features will occur in patients who have agenesis. This article describes a new approach that has been shown to minimize such a loss of anchorage. The technique is called hemisection and involves buccolingual sectioning of the second deciduous molar, followed by removal of its distal half. As necessary, the mesial half is also removed, and space closure can be completed. The advantages, philosophy, and technique are discussed in detail.