Literature DB >> 10190476

Oral-facial-digital syndrome with hypothalamic hamartoma, postaxial ray hypoplasia of the limbs, and vagino-cystic communication: a new variant?

I Fujiwara1, Y Kondo, K Iinuma.   

Abstract

We report on a 20-month-old girl with hypothalamic hamartoma, left cerebral atrophy, tongue nodules, oral frenula, micrognathia, hypoplasia of the left ulna, the fibulae, and right tibia, polysyndactyly of the hands and feet, vagino-cystic drainage with hydrometrocolpos, megaloureters, and hydronephrosis, agenesis of urethra, complex partial seizures, and central precocious puberty. The differential diagnosis is discussed. We conclude that the malformation complex in this girl is an oral-facial-digital syndrome, but is different from any of the 11 known subtypes.

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Year:  1999        PMID: 10190476

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


  2 in total

1.  Clinical-pathological conference: case 5.

Authors:  Craig B Fowler; Nisha J D'Silva
Journal:  Head Neck Pathol       Date:  2010-07-30

2.  Molecular analysis expands the spectrum of phenotypes associated with GLI3 mutations.

Authors:  Jennifer J Johnston; Julie C Sapp; Joyce T Turner; David Amor; Salim Aftimos; Kyrieckos A Aleck; Maureen Bocian; Joann N Bodurtha; Gerald F Cox; Cynthia J Curry; Ruth Day; Dian Donnai; Michael Field; Ikuma Fujiwara; Michael Gabbett; Moran Gal; John M Graham; Peter Hedera; Raoul C M Hennekam; Joseph H Hersh; Robert J Hopkin; Hülya Kayserili; Alexa M J Kidd; Virginia Kimonis; Angela E Lin; Sally Ann Lynch; Melissa Maisenbacher; Sahar Mansour; Julie McGaughran; Lakshmi Mehta; Helen Murphy; Margarita Raygada; Nathaniel H Robin; Alan F Rope; Kenneth N Rosenbaum; G Bradley Schaefer; Amy Shealy; Wendy Smith; Maria Soller; Annmarie Sommer; Heather J Stalker; Bernhard Steiner; Mark J Stephan; David Tilstra; Susan Tomkins; Pamela Trapane; Anne Chun-Hui Tsai; Margot I Van Allen; Pradeep C Vasudevan; Bernhard Zabel; Janice Zunich; Graeme C M Black; Leslie G Biesecker
Journal:  Hum Mutat       Date:  2010-10       Impact factor: 4.878

  2 in total

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