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
References (20)
20 references, click to expand
-
Subtle chromosomal rearrangements in children with unexplained mental retardation.
Lancet. 1999 Nov 13;354(9191):1676-81
PMID: 10568569
-
Screening for subtelomeric rearrangements in 210 patients with unexplained mental retardation using multiplex ligation dependent probe amplification (MLPA).
J Med Genet. 2004 Dec;41(12):892-9
PMID: 15591274
-
A 1.5 million-base pair inversion polymorphism in families with Williams-Beuren syndrome.
Nat Genet. 2001 Nov;29(3):321-5
PMID: 11685205
-
Physical map of 1p36, placement of breakpoints in monosomy 1p36, and clinical characterization of the syndrome.
Am J Hum Genet. 2003 May;72(5):1200-12
PMID: 12687501
-
The use of telomere probes to investigate submicroscopic rearrangements associated with mental retardation.
Curr Opin Genet Dev. 2003 Jun;13(3):310-6
PMID: 12787795
-
Telomeres: a diagnosis at the end of the chromosomes.
J Med Genet. 2003 Jun;40(6):385-98
PMID: 12807958
-
Implications of human genome architecture for rearrangement-based disorders: the genomic basis of disease.
Hum Mol Genet. 2004 Apr 1;13 Spec No 1:R57-64
PMID: 14764619
-
Molecular analysis of 20 patients with 2q37.3 monosomy: definition of minimum deletion intervals for key phenotypes.
J Med Genet. 2004 Jun;41(6):433-9
PMID: 15173228
-
Mutations in the DLG3 gene cause nonsyndromic X-linked mental retardation.
Am J Hum Genet. 2004 Aug;75(2):318-24
PMID: 15185169
-
Quaternary structure, protein dynamics, and synaptic function of SAP97 controlled by L27 domain interactions.
Neuron. 2004 Oct 28;44(3):453-67
PMID: 15504326
-
Deletions of chromosome 15 as a cause of the Prader-Willi syndrome.
N Engl J Med. 1981 Feb 5;304(6):325-9
PMID: 7442771
-
Detection of submicroscopic deletions in band 17p13 in patients with the Miller-Dieker syndrome.
Am J Hum Genet. 1988 Nov;43(5):597-604
PMID: 2903661
-
Angelman and Prader-Willi syndromes share a common chromosome 15 deletion but differ in parental origin of the deletion.
Am J Med Genet. 1989 Feb;32(2):285-90
PMID: 2564739
-
Hemizygosity at the elastin locus in a developmental disorder, Williams syndrome.
Nat Genet. 1993 Sep;5(1):11-6
PMID: 7693128
-
The detection of subtelomeric chromosomal rearrangements in idiopathic mental retardation.
Nat Genet. 1995 Feb;9(2):132-40
PMID: 7719339
-
A complete set of human telomeric probes and their clinical application. National Institutes of Health and Institute of Molecular Medicine collaboration.
Nat Genet. 1996 Sep;14(1):86-9
PMID: 8782825
-
Chromosome 1p36 deletions: the clinical phenotype and molecular characterization of a common newly delineated syndrome.
Am J Hum Genet. 1997 Sep;61(3):642-50
PMID: 9326330
-
PAK3 mutation in nonsyndromic X-linked mental retardation.
Nat Genet. 1998 Sep;20(1):25-30
PMID: 9731525
-
Screening for submicroscopic chromosome rearrangements in children with idiopathic mental retardation using microsatellite markers for the chromosome telomeres.
J Med Genet. 1999 May;36(5):405-11
PMID: 10353788
-
Cryptic telomeric rearrangements in subjects with mental retardation associated with dysmorphism and congenital malformations.
J Med Genet. 2001 Jun;38(6):417-20
PMID: 11424927