Literature DB >> 8588593

Familial microtia with meatal atresia and conductive deafness in five generations.

A Gupta1, M A Patton.   

Abstract

We describe a large family with congenital microtia, auditory meatal atresia and conductive deafness. The pedigree suggests autosomal dominant inheritance with variable expression and low penetrance. The literature is also reviewed to describe the inheritance pattern and clinical spectrum noted in this rare syndrome so far. The family is unique because the set of otologic anomalies in five generations was associated with renal cysts in one of the affected members, suggesting that this oto-renal (OR) syndrome may represent a variable expression of the branchio-oto-renal (BOR) syndrome. However, the probability is that this dominant malformation syndrome is a distinct entity.

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Year:  1995        PMID: 8588593     DOI: 10.1002/ajmg.1320590223

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


  6 in total

Review 1.  Genetic Advances in the Understanding of Microtia.

Authors:  Craig Gendron; Ann Schwentker; John A van Aalst
Journal:  J Pediatr Genet       Date:  2016-09-23

Review 2.  Microtia: epidemiology and genetics.

Authors:  Daniela V Luquetti; Carrie L Heike; Anne V Hing; Michael L Cunningham; Timothy C Cox
Journal:  Am J Med Genet A       Date:  2011-11-21       Impact factor: 2.802

3.  Microtia-anotia: a global review of prevalence rates.

Authors:  Daniela Varela Luquetti; Emanuele Leoncini; Pierpaolo Mastroiacovo
Journal:  Birth Defects Res A Clin Mol Teratol       Date:  2011-06-07

4.  A role of the double-stranded RNA-binding protein PACT in mouse ear development and hearing.

Authors:  Theresa M Rowe; Mark Rizzi; Keiko Hirose; Gregory A Peters; Ganes C Sen
Journal:  Proc Natl Acad Sci U S A       Date:  2006-03-29       Impact factor: 11.205

5.  Bilateral anotia with congenital hypothyroidism.

Authors:  Vaishali More; Sanjeev R Ahuja; Hemant V Kulkarni; Madhuri V Kulkarni
Journal:  Indian J Pediatr       Date:  2004-04       Impact factor: 1.967

6.  Sociodemographic and hispanic acculturation factors and isolated anotia/microtia.

Authors:  Adrienne T Hoyt; Mark A Canfield; Gary M Shaw; Dorothy K Waller; Kara N D Polen; Tunu Ramadhani; Marlene T Anderka; Angela E Scheuerle
Journal:  Birth Defects Res A Clin Mol Teratol       Date:  2014-07-30
  6 in total

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