Literature DB >> 9800904

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

E Horii1, Y Sugiura, R Nakamura.   

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.

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Year:  1998        PMID: 9800904     DOI: 10.1002/(sici)1096-8628(19981102)80:1<1::aid-ajmg1>3.0.co;2-8

Source DB:  PubMed          Journal:  Am J Med Genet        ISSN: 0148-7299


  5 in total

1.  Terminal osseous dysplasia with pigmentary defects maps to human chromosome Xq27.3-xqter.

Authors:  W Zhang; R Amir; D W Stockton; I B Van Den Veyver; C A Bacino; H Y Zoghbi
Journal:  Am J Hum Genet       Date:  2000-03-17       Impact factor: 11.025

2.  Terminal osseous dysplasia is caused by a single recurrent mutation in the FLNA gene.

Authors:  Yu Sun; Rowida Almomani; Emmelien Aten; Jacopo Celli; Jaap van der Heijden; Hanka Venselaar; Stephen P Robertson; Anna Baroncini; Brunella Franco; Lina Basel-Vanagaite; Emiko Horii; Ricardo Drut; Yavuz Ariyurek; Johan T den Dunnen; Martijn H Breuning
Journal:  Am J Hum Genet       Date:  2010-07-09       Impact factor: 11.025

3.  Diagnosis and treatment of digitocutaneous dysplasia, a rare infantile digital fibromatosis: a case report.

Authors:  Marisa Cabrera González; Laura M Pérez López; Diego Gutiérrez de la Iglesia; Carlota Rovira Zurriaga; Loreto Martorell Sampol; Antonia González Enseñat
Journal:  Hand (N Y)       Date:  2013-12

4.  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.

Authors:  Nicola Brunetti-Pierri; Ralph Lachman; Kwanghyuk Lee; Suzanne M Leal; Pasquale Piccolo; Ignatia B Van Den Veyver; Carlos A Bacino
Journal:  Am J Med Genet A       Date:  2010-07       Impact factor: 2.802

5.  Terminal osseous dysplasia with pigmentary defects and cardiomyopathy caused by a novel FLNA variant.

Authors:  Lynne Rumping; Marja W Wessels; Alex V Postma; Joost van Schuppen; Marjon A van Slegtenhorst; Jasper J Saris; J Peter van Tintelen; Stephen P Robertson; Mariëlle Alders; Saskia M Maas; Ronald H Lekanne Deprez
Journal:  Am J Med Genet A       Date:  2021-07-13       Impact factor: 2.578

  5 in total

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