Document Detail


Something to sink your teeth into.
MedLine Citation:
PMID:  21458016     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
A 21-year-old Kuwaiti man had cerebral palsy, retinitis pigmentosa, hypertension, and renal failure. His younger brother and sister displayed similar findings. Ocular motility examination disclosed aperiodic alternating skew deviation in the patient and his younger brother. Magnetic resonance imaging showed hypoplasia of the superior cerebellar vermis with a "molar tooth" sign. Genetic testing confirmed an AHPI gene mutation on chromosome 6p23.3 in the patient and his siblings, confirming the diagnosis of Joubert syndrome. This case demonstrates the need to consider the diagnosis of Joubert syndrome in adults with retinitis pigmentosa or familial alternating skew deviation.
Authors:
Steven M Couch; Scott E Brodie; Jacqueline A Leavitt; Michael C Brodsky
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Publication Detail:
Type:  Case Reports; Journal Article; Research Support, Non-U.S. Gov't     Date:  2011-04-01
Journal Detail:
Title:  Survey of ophthalmology     Volume:  56     ISSN:  1879-3304     ISO Abbreviation:  Surv Ophthalmol     Publication Date:    2011 Nov-Dec
Date Detail:
Created Date:  2011-11-28     Completed Date:  2012-01-27     Revised Date:  2012-03-22    
Medline Journal Info:
Nlm Unique ID:  0404551     Medline TA:  Surv Ophthalmol     Country:  United States    
Other Details:
Languages:  eng     Pagination:  544-9     Citation Subset:  IM    
Copyright Information:
Copyright © 2011 Elsevier Inc. All rights reserved.
Affiliation:
Department of Ophthalmology, Mayo Clinic, Rochester, Minnesota 55905, USA.
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MeSH Terms
Descriptor/Qualifier:
Adaptor Proteins, Signal Transducing / genetics
Cerebellar Diseases / diagnosis*,  genetics
Cerebellum / pathology*
Child
Child, Preschool
Chromosomes, Human, Pair 6 / genetics
Eye Abnormalities / diagnosis*,  genetics
Female
Humans
Hypertension / diagnosis,  genetics
Kidney Diseases, Cystic / diagnosis*,  genetics
Kidney Failure, Chronic / diagnosis,  genetics
Magnetic Resonance Imaging
Male
Mutation, Missense
Pedigree
Retina / abnormalities
Retinitis Pigmentosa / diagnosis,  genetics
Siblings
Visual Acuity
Young Adult
Chemical
Reg. No./Substance:
0/AHI1 protein, human; 0/Adaptor Proteins, Signal Transducing

From MEDLINE®/PubMed®, a database of the U.S. National Library of Medicine


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