Literature DB >> 1975860

Psychiatric findings in Wolfram syndrome homozygotes.

R G Swift1, D B Sadler, M Swift.   

Abstract

Diabetes mellitus and bilateral optic atrophy are the defining characteristics of the autosomal recessive Wolfram syndrome. Diabetes insipidus, neurogenic bladder, deafness, and other neurological manifestations are frequent. A review was made of the medical records of 68 Wolfram syndrome patients, aged between 8 and 43 years, identified by casefinding throughout the USA. 41 of the patients (60%) had episodes of severe depression, psychosis, or organic brain syndrome, as well as impulsive verbal and physical aggression. These symptoms were very severe in 17 patients (25%), of whom 12 required admission to a psychiatric hospital and 11 attempted suicide. We conclude that the Wolfram syndrome gene predisposes homozygotes to psychiatric illness.

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Year:  1990        PMID: 1975860     DOI: 10.1016/0140-6736(90)92157-d

Source DB:  PubMed          Journal:  Lancet        ISSN: 0140-6736            Impact factor:   79.321


  39 in total

1.  Mutations in the Wolfram syndrome 1 gene (WFS1) are a common cause of low frequency sensorineural hearing loss.

Authors:  I N Bespalova; G Van Camp; S J Bom; D J Brown; K Cryns; A T DeWan; A E Erson; K Flothmann; H P Kunst; P Kurnool; T A Sivakumaran; C W Cremers; S M Leal; M Burmeister; M M Lesperance
Journal:  Hum Mol Genet       Date:  2001-10-15       Impact factor: 6.150

2.  Mitochondrial abnormalities in the DIDMOAD syndrome.

Authors:  S Bundey; K Poulton; H Whitwell; E Curtis; I A Brown; A R Fielder
Journal:  J Inherit Metab Dis       Date:  1992       Impact factor: 4.982

3.  Impaired striatal dopamine output of homozygous Wfs1 mutant mice in response to [K+] challenge.

Authors:  Vallo Matto; Anton Terasmaa; Eero Vasar; Sulev Kõks
Journal:  J Physiol Biochem       Date:  2010-10-23       Impact factor: 4.158

4.  Wolfram syndrome: clinical and genetic profiling of a cohort from a tertiary care centre with characterization of the primary gonadal failure.

Authors:  Liza Das; Ashutosh Rai; Ravimohan Mavuduru; Kim Vaiphei; Akhilesh Sharma; Vishali Gupta; Sanjay Kumar Bhadada; Sailesh Lodha; Naresh Panda; Anil Bhansali; Paramjeet Singh; Pinaki Dutta
Journal:  Endocrine       Date:  2020-04-29       Impact factor: 3.633

5.  Autosomal recessive Wolfram syndrome associated with an 8.5-kb mtDNA single deletion.

Authors:  A Barrientos; J Casademont; A Saiz; F Cardellach; V Volpini; A Solans; E Tolosa; A Urbano-Marquez; X Estivill; V Nunes
Journal:  Am J Hum Genet       Date:  1996-05       Impact factor: 11.025

6.  Clinical and molecular assessment of 13 Iranian families with Wolfram syndrome.

Authors:  Maryam Sobhani; Mohammad Amin Tabatabaiefar; Soudeh Ghafouri-Fard; Asadollah Rajab; Sarah Mozafarpour; Samaneh Nasrniya; Abdol-Mohammad Kajbafzadeh; Mohammad Reza Noori-Daloii
Journal:  Endocrine       Date:  2019-07-16       Impact factor: 3.633

Review 7.  Psychiatric symptoms of inherited metabolic disease.

Authors:  Y Estrov; F Scaglia; O A Bodamer
Journal:  J Inherit Metab Dis       Date:  2000-02       Impact factor: 4.982

Review 8.  Inborn errors of metabolism associated with psychosis: literature review and case-control study using exome data from 5090 adult individuals.

Authors:  Yannis J Trakadis; Vanessa Fulginiti; Mark Walterfang
Journal:  J Inherit Metab Dis       Date:  2017-02-16       Impact factor: 4.982

9.  Neuropsychiatric Symptoms in Inborn Errors of Metabolism: Incorporation of Genomic and Metabolomic Analysis into Therapeutics and Prevention.

Authors:  Lisa Pan; Jerry Vockley
Journal:  Curr Genet Med Rep       Date:  2013-01-08

10.  Male mice with deleted Wolframin (Wfs1) gene have reduced fertility.

Authors:  Klari Noormets; Sulev Kõks; Ants Kavak; Andres Arend; Marina Aunapuu; Aivi Keldrimaa; Eero Vasar; Vallo Tillmann
Journal:  Reprod Biol Endocrinol       Date:  2009-08-10       Impact factor: 5.211

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