Dentigerous cyst involving both maxillary sinuses in a patient with hidrotic ectodermal dysplasia

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  • FUKUMOTO Chonji
    Department of Dentistry and Oral-Maxillofacial Surgery, Chiba University Hospital
  • KASAMATSU Atsushi
    Department of Dentistry and Oral-Maxillofacial Surgery, Chiba University Hospital
  • OGAWARA Katsunori
    Department of Dentistry and Oral-Maxillofacial Surgery, Chiba University Hospital
  • SHIIBA Masashi
    Department of Dentistry and Oral-Maxillofacial Surgery, Chiba University Hospital
  • UZAWA Katsuhiro
    Department of Dentistry and Oral-Maxillofacial Surgery, Chiba University Hospital
  • TANZAWA Hideki
    Department of Dentistry and Oral-Maxillofacial Surgery, Chiba University Hospital

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Other Title
  • 有汗性外胚葉異形成症患者に発生した両側上顎洞部含歯性嚢胞の1例
  • 有汗性外胚葉異形成症患者に発生した両側上顎洞部含歯性囊胞の1例
  • ユウカンセイ ガイハイヨウイケイセイショウ カンジャ ニ ハッセイ シタ リョウガワ ジョウガクドウブ ガン シセイノウホウ ノ 1レイ

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Abstract

Ectodermal dysplasia (ED), which is classified into three categories (anhidrotic, hypohidrotic, and hidrotic type), is a hereditary disease characerized by abnormal development of ectodermal tissues and organs. In the oral region, anodontia or oligodontia is the main feature of anhidrotic and hypohidrotic ED. However, intraoral symptoms in patients with hidrotic ED have not been reported. We describe a case of hidrotic ED, an uncommon subtype, associated with two large dentigerous cysts, which expanded the maxilla, eroded the maxillary sinuses, and projected into the nasal meatus. The present patient also had impacted teeth and retained deciduous teeth. The postoperative course was uneventful, and the patient was asymptomatic with no signs of recurrence 42 months postoperatively.

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