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A syndrome of digital fibromas, facial pigmentary dysplasia, and metacarpal and metatarsal disorganization.

作者信息

Horii E, Sugiura Y, Nakamura R

机构信息

Department of Orthopedics, Nagoya University School of Medicine, Japan.

出版信息

Am J Med Genet. 1998 Oct 30;80(1):1-5. doi: 10.1002/(sici)1096-8628(19981102)80:1<1::aid-ajmg1>3.0.co;2-8.

DOI:10.1002/(sici)1096-8628(19981102)80:1<1::aid-ajmg1>3.0.co;2-8
PMID:9800904
Abstract

We report on a girl with fibrous tumors involving hands and feet associated with unusual brachydactyly and facial abnormalities with pigmentary skin lesions. The multiple, infiltrative fibrous tumors clinically resembled recurring digital fibromata (RDF) of infancy, but eosinophilic cytoplasmic inclusion bodies pathognomonic for RDF were not demonstrated in her tumor cells. Skeletal alterations of the hands and feet comprised brachymesophalangy with cone-shaped epiphyses and variable shortening and deformity of the metacarpals and metatarsals. Facial abnormalities included upward slant of palpebral fissures, primary telecanthus with epicanthal folds, and a depressed nasal tip, along with a small fibrous tumor on the left eyelid. The patient also had patchy, brownish discolorations of the face. The clinical, radiological, and histological constellation in the patient may represent a hitherto undescribed hamartomatous syndrome.

摘要

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引用本文的文献

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Terminal osseous dysplasia with pigmentary defects and cardiomyopathy caused by a novel FLNA variant.一种新型 FLNA 变异导致的伴有色素缺陷和心肌病的终末骨发育不良。
Am J Med Genet A. 2021 Dec;185(12):3814-3820. doi: 10.1002/ajmg.a.62417. Epub 2021 Jul 13.
2
Diagnosis and treatment of digitocutaneous dysplasia, a rare infantile digital fibromatosis: a case report.指皮肤发育异常(一种罕见的婴儿期指纤维瘤病)的诊断与治疗:一例报告
Hand (N Y). 2013 Dec;8(4):473-8. doi: 10.1007/s11552-013-9515-8.
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Terminal osseous dysplasia is caused by a single recurrent mutation in the FLNA gene.
末端骨发育不良是由 FLNA 基因中的单个重复突变引起的。
Am J Hum Genet. 2010 Jul 9;87(1):146-53. doi: 10.1016/j.ajhg.2010.06.008.
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Terminal osseous dysplasia with pigmentary defects (TODPD): Follow-up of the first reported family, characterization of the radiological phenotype, and refinement of the linkage region.终端骨发育不良伴色素缺陷(TODPD):首例家族的随访、放射表型特征分析及连锁区域精确定位。
Am J Med Genet A. 2010 Jul;152A(7):1825-31. doi: 10.1002/ajmg.a.33470.
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Terminal osseous dysplasia with pigmentary defects maps to human chromosome Xq27.3-xqter.伴有色素沉着缺陷的终末骨发育异常定位于人类X染色体q27.3-qter区域。
Am J Hum Genet. 2000 Apr;66(4):1461-4. doi: 10.1086/302868. Epub 2000 Mar 17.