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PMID: 2199849 Published · ppublish English Case Reports Journal Article Research Support, Non-U.S. Gov't Research Support, U.S. Gov't, P.H.S.

Partial dystrophin deficiency in monozygous twin carriers of the Duchenne gene discordant for clinical myopathy.

Neurology ·Vol. 40 ·No. 8 ·1990-08-00 ·Pages 1267-70

Bonilla E, Younger DS, Chang HW, Tantravahi U, Miranda AF, Medori R, DiMauro S, Warburton D, Rowland LP

Abstract

We studied monozygous twin women, age 63. One, asymptomatic, had a serum creatine kinase (CK) level of 191 units (normal, 1 to 50); her son died of typical Duchenne muscular dystrophy (DMD) at age 18. Her twin sister had symptomatic limb weakness from about age 40. Her serum CK was 495 units. EMG and muscle biopsy were compatible with myopathy. In the asymptomatic twin, the peripheral blood lymphocyte karyotype was 46,XX. In the affected twin, 18% of cells were 45,X, and the others 46,XX, without X/autosome translocation. DNA analysis did not reveal a deletion at the DMD locus. Immunologic studies of dystrophin showed a partial deficiency of the protein that was more severe in the symptomatic twin. The clinical discordance and the different severity of dystrophin deficiency may have resulted from the effects of lyonization.

MeSH Terms
Creatine Kinase/blood Diseases in Twins Dystrophin Female Genetic Carrier Screening Humans Male Middle Aged Muscle Proteins/analysis,deficiency,genetics Muscles/cytology,pathology Muscular Dystrophies/genetics Sarcolemma/ultrastructure Twins, Monozygotic
Chemicals
Dystrophin Muscle Proteins Creatine Kinase
Authors & Affiliations
9 authors, click to expand affiliations / ORCID
Bonilla E
Department of Neurology, Columbia University, College of Physicians and Surgeons, New York, NY 10032.
Younger D S
Chang H W
Tantravahi U
Miranda A F
Medori R
DiMauro S
Warburton D
Rowland L P
Article Info
Journal
Neurology
Abbr.
Neurology
ISSN
0028-3878
Published
1990-08-00
Pages
1267-70
Language
English
Region
United States
NLM ID
0401060
Subset
IM
Grants
NINDS NIH HHS · NS-11766 · United States
Analysis Services
Analysis Services

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