Literature DB >> 8256813

Persistence of müllerian derivatives, lymphangiectasis, hepatic failure, postaxial polydactyly, renal and craniofacial anomalies.

M Urioste1, J I Rodríguez, J M Barcia, M Martín, R Escribá, M Pardo, J Camino, M L Martínez-Frías.   

Abstract

We describe 3 unrelated newborn males with a previously unreported constellation of congenital anomalies. All 3 died neonatally of hepatic failure. Clinically, they presented with a pattern of malformations characterized by prenatal linear growth deficiency, hypertrophied alveolar ridges, redundant nuchal skin, and postaxial polydactyly. All 3 cases had male external genitalia with cryptorchidism, and 2 of them, a small penis. Necropsies showed similar internal anomalies, consisting of müllerian duct remnants, lymphangiectasis, and renal anomalies. The karyotypes were normal (46, XY) in skin fibroblasts (Case 1) and in peripheral blood lymphocytes (Case 3). Although this pattern of congenital anomalies must be differentiated from several other lethal syndromes, to our knowledge, no similar cases have been described previously. Cause of this syndrome is unknown. Because Case 2 had a previous brother with similar anomalies, we suspect that this new entity probably is an autosomal recessive or X-linked trait.

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Year:  1993        PMID: 8256813     DOI: 10.1002/ajmg.1320470413

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


  4 in total

Review 1.  Molecular genetics of Müllerian duct formation, regression and differentiation.

Authors:  Rachel D Mullen; Richard R Behringer
Journal:  Sex Dev       Date:  2014-07-12       Impact factor: 1.824

Review 2.  Ocular lymphatics: state-of-the-art review.

Authors:  L Chen
Journal:  Lymphology       Date:  2009-06       Impact factor: 1.286

Review 3.  A novel CCBE1 mutation leading to a mild form of hennekam syndrome: case report and review of the literature.

Authors:  Patrick Frosk; Bernard Chodirker; Louise Simard; Wael El-Matary; Ana Hanlon-Dearman; Jeremy Schwartzentruber; Jacek Majewski; Cheryl Rockman-Greenberg
Journal:  BMC Med Genet       Date:  2015-04-30       Impact factor: 2.103

Review 4.  Congenital pulmonary lymphangiectasia.

Authors:  Carlo Bellini; Francesco Boccardo; Corradino Campisi; Eugenio Bonioli
Journal:  Orphanet J Rare Dis       Date:  2006-10-30       Impact factor: 4.123

  4 in total

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