Literature DB >> 7246619

Nonprogressive cerebellar disorder with mental retardation and autosomal recessive inheritance in Hutterites.

V Schurig, A V Orman, P Bowen.   

Abstract

We report a nonprogressive neurological disorder in at least 11 Hutterites with healthy but consanguineous parents. In several of the affected, hypotonia was noted at birth. Retarded motor and mental development became apparent during the first year of life. The age of unsupported walking varied from 5-21 years. Consistent signs were unsteady, broadly based gait and stance, exaggerated deep tendon reflexes mainly in the lower limbs, and mild to moderate mental retardation. Variable signs were extensor plantar reflexes (9/11), short stature (-2SD in 8/11), strabismus (7/11), small muscle mass (6/11), mild intention tremor (3/11), cataracts (1/11), and epilepsy (1/11). CAT scans in two affected sisters showed slight enlargement of the fourth ventricle in one and hypoplasia of the cerebellum in both. The disorder is probably the same as that described earlier under the heading, dysequilibrium syndrome.

Entities:  

Mesh:

Year:  1981        PMID: 7246619     DOI: 10.1002/ajmg.1320090109

Source DB:  PubMed          Journal:  Am J Med Genet        ISSN: 0148-7299


  11 in total

1.  Reply to Herz et al. and Humphrey et al.: Genetic heterogeneity of cerebellar hypoplasia with quadrupedal locomotion.

Authors:  Tayfun Ozcelik; Nurten Akarsu; Elif Uz; Safak Caglayan; Suleyman Gulsuner; Onur Emre Onat; Meliha Tan; Uner Tan
Journal:  Proc Natl Acad Sci U S A       Date:  2008-06-10       Impact factor: 11.205

2.  Cerebellar ataxia, mental retardation and dysequilibrium syndrome 1 (CAMRQ1) caused by an unusual constellation of VLDLR mutation.

Authors:  Lars Schlotawa; Alrun Hotz; Christine Zeschnigk; Britta Hartmann; Jutta Gärtner; Deborah Morris-Rosendahl
Journal:  J Neurol       Date:  2013-05-14       Impact factor: 4.849

3.  Homozygous deletion of the very low density lipoprotein receptor gene causes autosomal recessive cerebellar hypoplasia with cerebral gyral simplification.

Authors:  Kym M Boycott; Shauna Flavelle; Alexandre Bureau; Hannah C Glass; T Mary Fujiwara; Elaine Wirrell; Krista Davey; Albert E Chudley; James N Scott; D Ross McLeod; Jillian S Parboosingh
Journal:  Am J Hum Genet       Date:  2005-07-22       Impact factor: 11.025

4.  Clinical correlation of dysequilibrium syndrome and 4-hydroxybutyric aciduria.

Authors:  K M Gibson; L Sweetman; W L Nyhan; P Bowen
Journal:  J Inherit Metab Dis       Date:  1985       Impact factor: 4.982

5.  Novel VLDLR microdeletion identified in two Turkish siblings with pachygyria and pontocerebellar atrophy.

Authors:  Luis E Kolb; Zulfikar Arlier; Cengiz Yalcinkaya; Ali K Ozturk; Jennifer A Moliterno; Ozdem Erturk; Fatih Bayrakli; Baris Korkmaz; Michael L DiLuna; Katsuhito Yasuno; Kaya Bilguvar; Tayfun Ozcelik; Beyhan Tuysuz; Matthew W State; Murat Gunel
Journal:  Neurogenetics       Date:  2010-01-15       Impact factor: 2.660

6.  Very mild features of dysequilibrium syndrome associated with a novel VLDLR missense mutation.

Authors:  Alessia Micalizzi; Isabella Moroni; Monia Ginevrino; Tommaso Biagini; Tommaso Mazza; Marta Romani; Enza Maria Valente
Journal:  Neurogenetics       Date:  2016-06-02       Impact factor: 2.660

7.  Mutations in VLDLR as a cause for autosomal recessive cerebellar ataxia with mental retardation (dysequilibrium syndrome).

Authors:  Kym M Boycott; Carsten Bonnemann; Joachim Herz; Stephanie Neuert; Chandree Beaulieu; James N Scott; Anuradha Venkatasubramanian; Jillian S Parboosingh
Journal:  J Child Neurol       Date:  2009-03-30       Impact factor: 1.987

8.  Midbrain-hindbrain involvement in lissencephalies.

Authors:  Patrice Jissendi-Tchofo; Simay Kara; A James Barkovich
Journal:  Neurology       Date:  2008-11-19       Impact factor: 9.910

9.  Mutation in NSUN2, which encodes an RNA methyltransferase, causes autosomal-recessive intellectual disability.

Authors:  Muzammil Ahmad Khan; Muhammad Arshad Rafiq; Abdul Noor; Shobbir Hussain; Joana V Flores; Verena Rupp; Akshita K Vincent; Roland Malli; Ghazanfar Ali; Falak Sher Khan; Gisele E Ishak; Dan Doherty; Rosanna Weksberg; Muhammad Ayub; Christian Windpassinger; Shahnaz Ibrahim; Michaela Frye; Muhammad Ansar; John B Vincent
Journal:  Am J Hum Genet       Date:  2012-04-26       Impact factor: 11.025

10.  A missense founder mutation in VLDLR is associated with Dysequilibrium Syndrome without quadrupedal locomotion.

Authors:  Bassam R Ali; Jennifer L Silhavy; Matthew J Gleeson; Joseph G Gleeson; Lihadh Al-Gazali
Journal:  BMC Med Genet       Date:  2012-09-14       Impact factor: 2.103

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