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Automated comparative sequence analysis identifies mutations in 89% of NF1 patients and confirms a mutation cluster in exons 11-17 distinct from the GAP related domain4

Automated comparative sequence analysis identifies mutations in 89% of NF1 patients and confirms a mutation cluster in exons 11-17 distinct from the GAP related domain4
Automated comparative sequence analysis identifies mutations in 89% of NF1 patients and confirms a mutation cluster in exons 11-17 distinct from the GAP related domain4
Neurofibromatosis type 1 (NF1), formerly known as Von Recklinghausen Neurofibromatosis, is a common genetic disorder affecting approximately 1 in 3000–5000 people. It is a fully penetrant autosomal dominant disorder. Strict diagnostic criteria that include café au lait spots, neurofibromas, plexiform neurofibromas, freckling in the axillary or inguinal regions, Lisch nodules (iris haematomas), optic or chiasma glioma, pseudoarthrosis, and sphenoid dysplasia define NF1. Most disease features are present in more than 90% of patients at puberty.1 Further manifestations are known to occur in this disorder, including macrocephaly, short stature, learning difficulties, scoliosis and certain malignancies.2–4 There is, however, great intra and interfamilial phenotypic variability. In addition a number of patients who have a clinical picture suspected to be NF1 do not fulfil the diagnostic criteria particularly in the younger age groups. As a consequence genetic testing would have a major impact on the diagnosis and management of these families.
research, sequence analysis, chemistry, methods, comparative study, humans, neurofibromatosis 1, polymorphism, proteins, neurofibromin 1, diagnosis, dna mutational analysis, genes, tertiary, mutation, protein structure, genetics, protein, analysis, genetic, exons, research support, patients, evaluation studies, gtpase-activating proteins
0022-2593
e48
Mattocks, C.
d9220649-2064-473f-b004-0895bf9fec97
Baralle, D.
faac16e5-7928-4801-9811-8b3a9ea4bb91
Tarpey, P.
e1d6a030-5081-41b1-945c-c6744749999d
Ffrench-Constant, C.
d9c008e9-ae0f-4664-adcd-c32abf95381d
Bobrow, M.
dd69aef7-6b9e-4f90-b2b5-611d7f57c2cd
Whittaker, J.
fbd09856-ad0d-44e4-9a33-3a904b70c0bc
Mattocks, C.
d9220649-2064-473f-b004-0895bf9fec97
Baralle, D.
faac16e5-7928-4801-9811-8b3a9ea4bb91
Tarpey, P.
e1d6a030-5081-41b1-945c-c6744749999d
Ffrench-Constant, C.
d9c008e9-ae0f-4664-adcd-c32abf95381d
Bobrow, M.
dd69aef7-6b9e-4f90-b2b5-611d7f57c2cd
Whittaker, J.
fbd09856-ad0d-44e4-9a33-3a904b70c0bc

Mattocks, C., Baralle, D., Tarpey, P., Ffrench-Constant, C., Bobrow, M. and Whittaker, J. (2004) Automated comparative sequence analysis identifies mutations in 89% of NF1 patients and confirms a mutation cluster in exons 11-17 distinct from the GAP related domain4. Journal of Medical Genetics, 41 (4), e48.

Record type: Article

Abstract

Neurofibromatosis type 1 (NF1), formerly known as Von Recklinghausen Neurofibromatosis, is a common genetic disorder affecting approximately 1 in 3000–5000 people. It is a fully penetrant autosomal dominant disorder. Strict diagnostic criteria that include café au lait spots, neurofibromas, plexiform neurofibromas, freckling in the axillary or inguinal regions, Lisch nodules (iris haematomas), optic or chiasma glioma, pseudoarthrosis, and sphenoid dysplasia define NF1. Most disease features are present in more than 90% of patients at puberty.1 Further manifestations are known to occur in this disorder, including macrocephaly, short stature, learning difficulties, scoliosis and certain malignancies.2–4 There is, however, great intra and interfamilial phenotypic variability. In addition a number of patients who have a clinical picture suspected to be NF1 do not fulfil the diagnostic criteria particularly in the younger age groups. As a consequence genetic testing would have a major impact on the diagnosis and management of these families.

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More information

Published date: 2004
Keywords: research, sequence analysis, chemistry, methods, comparative study, humans, neurofibromatosis 1, polymorphism, proteins, neurofibromin 1, diagnosis, dna mutational analysis, genes, tertiary, mutation, protein structure, genetics, protein, analysis, genetic, exons, research support, patients, evaluation studies, gtpase-activating proteins

Identifiers

Local EPrints ID: 60037
URI: http://eprints.soton.ac.uk/id/eprint/60037
ISSN: 0022-2593
PURE UUID: b0152061-d564-428a-931d-a43b12840ddc
ORCID for D. Baralle: ORCID iD orcid.org/0000-0003-3217-4833

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Date deposited: 04 Sep 2008
Last modified: 12 Dec 2021 03:40

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Contributors

Author: C. Mattocks
Author: D. Baralle ORCID iD
Author: P. Tarpey
Author: C. Ffrench-Constant
Author: M. Bobrow
Author: J. Whittaker

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