Evidence map›Paper›PMID 24167463›Full record

ArticleMolecular syndromology2013

Interstitial deletions at 6q14.1q15 associated with developmental delay and a marfanoid phenotype.

R B Lowry, J E Chernos, M S Connelly, J P H Wyse

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In one paragraph

Article in Molecular syndromology, 2013. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 4 papers.

0numbers the graph read from it
0cells of the map it votes in
4citing papers in PubMed
–field-weighted citation impact
1 · What the graph read from it

What it found

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2 · The registry

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3 · Its place in the literature

Who cites it

4 citing papers in PubMed.

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4 · The record

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5 · Who and what money

Authors and funding

4 authors.

R B LowryDepartment of Medical Genetics, University of Calgary, Alta., Canada ; Department of Pediatrics, University of Calgary, Alta., Canada ; Department of Alberta Children's Hospital Research Institute, Calgary, Alta., Canada.
J E Chernos
M S Connelly
J P H Wyse

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

There are a number of reports of interstitial deletions of the long arm of chromosome 6 that have developmental delay and obesity suggesting that this is a distinct phenotype almost like Prader-Willi syndrome. Here we report a patient with a similar deletion but a strikingly different phenotype, one more in keeping with Marfan syndrome, although he does not fulfil the criteria for that syndrome. Array comparative genomic hybridization was performed to investigate a patient with a striking phenotype. This revealed an interstitial deletion of 6q14.1q15. Parental FISH studies were normal, indicating that this is a de novo deletion. Our patient has a completely different phenotype compared to other patients reported to have similar deletions. The common feature is developmental delay, but the body features are quite different in that our patient is tall, strikingly thin with pectus excavatum, scoliosis, skin striae, arachnodactyly, pes planus, cataracts, and a high-arched palate. This contrasts with other patients who have a similar deletion but have short stature and obesity. 6q14.1q15 interstitial deletions can have a very variable phenotype and do not necessarily conform to a clinical recognizable microdeletion syndrome caused by haploinsufficiency of dosage-sensitive genes in that region as proposed by others.

Indexed as

6q deletionDevelopmental delayMarfanoid phenotypeMental retardationTall stature

Identifiers

PMID24167463
PMCPMC3776401

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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.