Document Detail


Mardini-Nyhan association (lung agenesis, congenital heart, and thumb anomalies): three new cases and possible recurrence in a sib-is there a distinct recessive syndrome?
MedLine Citation:
PMID:  19938090     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
In 1985, Mardini and Nyhan described three patients from consanguineous families with unilateral complete/partial lung agenesis, congenital cardiac defects, and ipsilateral thumb anomalies. Although there have been many reports of lung agenesis with other malformations, especially hemifacial microsomia and radial ray anomalies, very few demonstrate this triad of defects. We describe three patients with the Mardini-Nyhan association which may represent a distinct entity, although this remains uncertain at present. A fourth patient is also described, the sister of one of the other patients, with complex congenital cardiac disease and bilateral lung lobation abnormalities. This is the first reported incidence of a possible recurrence within a family and suggests, together with the consanguinity observed by Mardini and Nyhan, that recessive inheritance should be considered in genetic counseling for this disorder.
Authors:
Rob Hastings; David Harding; Alan Donaldson; Rachel Liebling; Alison Hayes; Alison Kraus; Shelagh Joss; Shuba Narayanaswamy; Peter Turnpenny; Sarah Smithson
Publication Detail:
Type:  Case Reports; Journal Article    
Journal Detail:
Title:  American journal of medical genetics. Part A     Volume:  149A     ISSN:  1552-4833     ISO Abbreviation:  Am. J. Med. Genet. A     Publication Date:  2009 Dec 
Date Detail:
Created Date:  2009-11-30     Completed Date:  2010-01-27     Revised Date:  -    
Medline Journal Info:
Nlm Unique ID:  101235741     Medline TA:  Am J Med Genet A     Country:  United States    
Other Details:
Languages:  eng     Pagination:  2838-42     Citation Subset:  IM    
Affiliation:
Department of Clinical Genetics, University Hospitals Bristol NHS Foundation Trust, Bristol, UK.
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MeSH Terms
Descriptor/Qualifier:
Abnormalities, Multiple / pathology*
Fatal Outcome
Female
Heart Defects, Congenital / complications*
Humans
Infant
Infant, Newborn
Lung / abnormalities*,  radiography
Magnetic Resonance Imaging
Pregnancy
Recurrence
Siblings*
Syndrome
Thumb / abnormalities*,  radiography
Tomography, X-Ray Computed

From MEDLINE®/PubMed®, a database of the U.S. National Library of Medicine


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