Chondrodysplasia-disorder of sex development syndrome

Chondrodysplasia - disorder of sex development is an extremely rare disorder of sex development (see this term), reported in only two siblings (one terminated in pregnancy) to date, characterized by the clinical features of 46,XY complete gonadal dysgenesis (see this term; normal external female genitalia, lack of pubertal development, primary amenorrhea, and hypergonadotrophic hypogonadism) in association with severe dwarfism with generalized chondrodysplasia (bell-shaped thorax, micromelia, brachydactyly). Other reported features in the live sibling included eye anomalies (hypoplastic irides, myopia, coloboma of optic discs), dysmorphic features (deep-set eyes, upslanting palpebral fissures, puffy eyelids, large ears and mouth, mild prognathism), muscular hypoplasia, mild intellectual deficiency and severe microcephaly with cerebellar vermis hypoplasia. An autosomal recessive inheritance has been suggested.



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症例報告を絞り込む



合計: 1 (症例報告)

  


(表示件数)
対応する徴候・症状  遺伝子  変異  キーワード(MeSH)
順位
(類似度)
PMID
(PMCID)
1
(39.0%)
30912300
Biallelic novel missense HHAT variant causes syndromic microcephaly and cerebellar-vermis hypoplasia.
Abdel-Salam GMH, Mazen I, Eid M, Ewida N, Shaheen R, Alkuraya FS.
Am J Med Genet A. 2019;179(6):1053-1057.
小頭
HHAT
c|SUB|T|770|C;RS#:780669166 p|SUB|L|257|P;RS#:761454249
DNA変異解析 アシル基転移酵素 ヒト ミスセンス変異 子供 子供(未就学) 対立遺伝子 小頭症 新生児 疾患の遺伝的素因 磁気共鳴画像法 表現型 遺伝子関連解析 顔貌
        

徴候・症状リスト(Orphanetデータベースから取得)

    合計: 22

HPO ID 徴候・症状 頻度
HP:0000252 小頭 Very frequent (99-80%)
HP:0000400 大耳 Very frequent (99-80%)
HP:0000506 内眼角外方偏位 Very frequent (99-80%)
HP:0000774 狭い胸郭 Very frequent (99-80%)
HP:0001249 知的障害 Very frequent (99-80%)
HP:0001511 子宮内成長遅滞 Very frequent (99-80%)
HP:0002644 骨盤帯骨の形態異常 Very frequent (99-80%)
HP:0002983 小肢症 Very frequent (99-80%)
HP:0003043 肩の異常 Very frequent (99-80%)
HP:0003510 重度の低身長 Very frequent (99-80%)
HP:0004330 頭蓋骨骨化増加 Very frequent (99-80%)
HP:0005622 幅広い長管骨 Very frequent (99-80%)
HP:0009803 短い指骨 Very frequent (99-80%)
HP:0010049 短い中手骨 Very frequent (99-80%)
HP:0000037 男性仮性半陰陽 Frequent (79-30%)
HP:0000486 斜視 Frequent (79-30%)
HP:0000490 落ちくぼんだ眼 Frequent (79-30%)
HP:0000567 脈絡膜網膜コロボーマ Frequent (79-30%)
HP:0000581 眼瞼裂狭小 Frequent (79-30%)
HP:0000616 縮瞳 Frequent (79-30%)
HP:0001322 非常に小さな脳 Frequent (79-30%)
HP:0007676 虹彩低形成 Frequent (79-30%)


徴候・症状リスト(症例報告から取得)

    合計: 0

HPO ID 徴候・症状 症例報告数


疾患原因遺伝子リスト(Orphanetデータベースから取得)

    合計: 1

Gene Symbol 遺伝子名 Entrez Gene ID
HHAT hedgehog acyltransferase 55733