Abstract
We have developed a fast and accurate PCR-based linkage and carrier detection protocol for families of Duchenne muscular dystrophy (DMD)/Becker muscular dystrophy (BMD) patients with or without detectable deletions of the dystrophin gene, using fluorescent PCR products analyzed on an automated sequencer. When a deletion is found in the affected male DMD/BMD patient by standard multiplex PCR, fluorescently labeled primers specific for the deleted and nondeleted exon(s) are used to amplify the DNA of at-risk female relatives by using multiplex PCR at low cycle number (20 cycles). The products are then quantitatively analyzed on an automatic sequencer to determine whether they are heterozygous for the deletion and thus are carriers. As a confirmation of the deletion data, and in cases in which a deletion is not found in the proband, fluorescent multiplex PCR linkage is done by using four previously described polymorphic dinucleotide sequences. The four (CA)n repeats are located throughout the dystrophin gene, making the analysis highly informative and accurate. We present the successful application of this protocol in families who proved refractory to more traditional analyses.
MeSH Terms
DNA/blood,genetics,isolation & purification
Dystrophin/genetics
Exons
Female
Gene Deletion
Genetic Carrier Screening
Genetic Linkage
Humans
Male
Muscular Dystrophies/diagnosis,genetics
Oligodeoxyribonucleotides
Pedigree
Polymerase Chain Reaction
Pregnancy
Prenatal Diagnosis
Spectrometry, Fluorescence
Chemicals
Dystrophin
Oligodeoxyribonucleotides
DNA
Authors & Affiliations
5 authors, click to expand affiliations / ORCID
Schwartz L S
Department of Molecular Genetics, University of Pittsburgh School of Medicine, PA 15261.
Tarleton J
Popovich B
Seltzer W K
Hoffman E P
References (18)
18 references, click to expand
-
Identification of a nondeletion defect in alpha-thalassemia.
N Engl J Med. 1977 Nov 17;297(20):1081-4
PMID: 909565
-
Complete cloning of the Duchenne muscular dystrophy (DMD) cDNA and preliminary genomic organization of the DMD gene in normal and affected individuals.
Cell. 1987 Jul 31;50(3):509-17
PMID: 3607877
-
A polymorphic CACA repeat in the 3' untranslated region of dystrophin.
Nucleic Acids Res. 1990 Apr 11;18(7):1931
PMID: 1970874
-
Detection of 98% of DMD/BMD gene deletions by polymerase chain reaction.
Hum Genet. 1990 Nov;86(1):45-8
PMID: 2253937
-
Carrier detection and prenatal diagnosis in Duchenne and Becker muscular dystrophy.
Br Med Bull. 1989 Jul;45(3):719-44
PMID: 2688825
-
Prenatal diagnosis and carrier detection of Duchenne muscular dystrophy with closely linked RFLPs.
Lancet. 1985 Mar 23;1(8430):655-8
PMID: 2858615
-
Dystrophin: the protein product of the Duchenne muscular dystrophy locus.
Cell. 1987 Dec 24;51(6):919-28
PMID: 3319190
-
Prediction of carrier status in Duchenne dystrophy by creatine kinase measurement.
Am J Clin Pathol. 1985 Nov;84(5):655-8
PMID: 4061390
-
Dystrophin and disease.
Mol Aspects Med. 1991;12(3):175-94
PMID: 1770836
-
A convenient multiplex PCR system for the detection of dystrophin gene deletions: a comparative analysis with cDNA hybridisation shows mistypings by both methods.
J Med Genet. 1991 May;28(5):304-11
PMID: 1865467
-
An informative polymorphism detectable by polymerase chain reaction at the 3' end of the dystrophin gene.
Hum Genet. 1990 Feb;84(3):283-5
PMID: 1968037
-
Rapid detection of CA polymorphisms in cloned DNA: application to the 5' region of the dystrophin gene.
Am J Hum Genet. 1991 Mar;48(3):621-7
PMID: 1998344
-
Duplicational mutation at the Duchenne muscular dystrophy locus: its frequency, distribution, origin, and phenotypegenotype correlation.
Am J Hum Genet. 1990 Apr;46(4):682-95
PMID: 2316519
-
Correlation of clinical and deletion data in Duchenne and Becker muscular dystrophy.
J Med Genet. 1989 Nov;26(11):682-93
PMID: 2585468
-
Dystrophin abnormalities in Duchenne/Becker muscular dystrophy.
Neuron. 1989 Jan;2(1):1019-29
PMID: 2696500
-
Abundant class of human DNA polymorphisms which can be typed using the polymerase chain reaction.
Am J Hum Genet. 1989 Mar;44(3):388-96
PMID: 2916582
-
A simple salting out procedure for extracting DNA from human nucleated cells.
Nucleic Acids Res. 1988 Feb 11;16(3):1215
PMID: 3344216
-
Carrier detection and prenatal diagnosis in Duchenne and Becker muscular dystrophy families, using dinucleotide repeat polymorphisms.
Am J Hum Genet. 1991 Nov;49(5):951-60
PMID: 1928100