Literature DB >> 19938094

Overlap of Moebius and oromandibular limb hypogenesis syndrome with gastroschisis and pulmonary hypoplasia.

Knut Brockmann1, Heiko Backes, Bernd Auber, Thomas Kriebel, Franziska Stellmer, Barbara Zoll.   

Abstract

The oromandibular limb hypogenesis syndromes (OLHS) represent a group of rare conditions characterized by congenital malformations involving the tongue, mandible, and limbs. There is considerable overlap between the syndromes gathered under the term OLHS, and a marked variability of face and limb anomalies as well as additional malformations. In this report we describe a girl with gastroschisis and pulmonary hypoplasia in addition to features of Moebius syndrome comprising hypoplasia of the tongue and mandible, brachydactyly of halluces, cranial nerve palsies with bilateral facial paralysis and an inability to execute horizontal eye movements. Karyotyping and array-based comparative genomic hybridization were normal. This observation confirms an overlap between Moebius syndrome and OLHS and widens the spectrum of associated malformations. Intrauterine environmental factors including vascular insufficiency, high maternal fever, and drug abuse are likely to play a crucial role in the pathogenesis of this condition.

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Year:  2009        PMID: 19938094     DOI: 10.1002/ajmg.a.33111

Source DB:  PubMed          Journal:  Am J Med Genet A        ISSN: 1552-4825            Impact factor:   2.802


  2 in total

Review 1.  A clinical-pathogenetic approach on associated anomalies and chromosomal defects supports novel candidate critical regions and genes for gastroschisis.

Authors:  Victor M Salinas-Torres; Rafael A Salinas-Torres; Ricardo M Cerda-Flores; Hugo L Gallardo-Blanco; Laura E Martínez-de-Villarreal
Journal:  Pediatr Surg Int       Date:  2018-08-09       Impact factor: 1.827

2.  Oromandibular-limb Hypogenesis Syndrome Type II C: A Rare Case.

Authors:  Renita Lorina Castelino; Shishir Ram Shetty; Subhas Babu G; Kumuda Arvind Rao H T
Journal:  J Dent Res Dent Clin Dent Prospects       Date:  2010-12-21
  2 in total

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