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PMID: 15918153 Published · ppublish English Journal Article Research Support, Non-U.S. Gov't

3q29 microdeletion syndrome: clinical and molecular characterization of a new syndrome.

American journal of human genetics ·Vol. 77 ·No. 1 ·2005-07-00 ·Pages 154-60

Willatt L, Cox J, Barber J, Cabanas ED, Collins A, Donnai D, FitzPatrick DR, Maher E, Martin H, Parnau J, Pindar L, Ramsay J, Shaw-Smith C, Sistermans EA, Tettenborn M, Trump D, de Vries BB, Walker K, Raymond FL

Abstract

We report the identification of six patients with 3q29 microdeletion syndrome. The clinical phenotype is variable despite an almost identical deletion size. The phenotype includes mild-to-moderate mental retardation, with only slightly dysmorphic facial features that are similar in most patients: a long and narrow face, short philtrum, and high nasal bridge. Autism, gait ataxia, chest-wall deformity, and long and tapering fingers were noted in at least two of six patients. Additional features--including microcephaly, cleft lip and palate, horseshoe kidney and hypospadias, ligamentous laxity, recurrent middle ear infections, and abnormal pigmentation--were observed, but each feature was only found once, in a single patient. The microdeletion is approximately 1.5 Mb in length, with molecular boundaries mapping within the same or adjacent bacterial artificial chromosome (BAC) clones at either end of the deletion in all patients. The deletion encompasses 22 genes, including PAK2 and DLG1, which are autosomal homologues of two known X-linked mental retardation genes, PAK3 and DLG3. The presence of two nearly identical low-copy repeat sequences in BAC clones on each side of the deletion breakpoint suggests that nonallelic homologous recombination is the likely mechanism of disease causation in this syndrome.

MeSH Terms
Abnormalities, Multiple/genetics Child Child, Preschool Chromosome Deletion Chromosome Mapping Chromosomes, Human, Pair 3 Craniofacial Abnormalities/genetics Female Humans Infant Intellectual Disability/genetics Male Phenotype Syndrome
Authors & Affiliations
19 authors, click to expand affiliations / ORCID
Willatt Lionel
Department of Medical Genetics, Addenbrooke's Hospital, University of Cambridge, Cambridge, United Kingdom.
Cox James
Barber John
Cabanas Elisabet Dachs
Collins Amanda
Donnai Dian
FitzPatrick David R
Maher Eddy
Martin Howard
Parnau Josep
Pindar Lesley
Ramsay Jacqueline
Shaw-Smith Charles
Sistermans Erik A
Tettenborn Michael
Trump Dorothy
de Vries Bert B A
Walker Kate
Raymond F Lucy
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Article Info
Journal
American journal of human genetics
Abbr.
Am J Hum Genet
ISSN
0002-9297
Published
2005-07-00
Epub
2005-00-25
Pages
154-60
Language
English
Region
United States
NLM ID
0370475
PMCID
PMC1226188
Subset
IM
Grants
Wellcome Trust · United Kingdom
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