Literature DB >> 19767415

Prevalence of genetic muscle disease in Northern England: in-depth analysis of a muscle clinic population.

Fiona L M Norwood1, Chris Harling, Patrick F Chinnery, Michelle Eagle, Kate Bushby, Volker Straub.   

Abstract

We have performed a detailed population study of patients with genetic muscle disease in the northern region of England. Our current clinic population comprises over 1100 patients in whom we have molecularly characterized 31 separate muscle disease entities. Diagnostic clarity achieved through careful delineation of clinical features supported by histological, immunological and genetic analysis has allowed us to reach a definitive diagnosis in 75.7% of our patients. We have compared our case profile with that from Walton and Nattrass' seminal study from 1954, also of the northern region, together with data from other more recent studies from around the world. Point prevalence figures for each of the five major disease categories are comparable with those from other recent studies. Myotonic dystrophies are the most common, comprising 28.6% of our clinic population with a point prevalence of 10.6/100,000. Next most frequent are the dystrophinopathies and facioscapulohumeral muscular dystrophy making up 22.9% (8.46/100,000) and 10.7% (3.95/100,000) of the clinic population, respectively. Spinal muscular atrophy patients account for 5.1% or 1.87/100,000 patients. Limb girdle muscular dystrophy, which was described for the first time in the paper by Walton and Nattrass (1954) and comprised 17% of their clinic population, comprises 6.2% of our clinic population at a combined prevalence of 2.27/100,000. The clinic population included patients with 12 other muscle disorders. These disorders ranged from a point prevalence of 0.89/100 000 for the group of congenital muscular dystrophies to conditions with only two affected individuals in a population of three million. For the first time our study provides epidemiological information for X-linked Emery-Dreifuss muscular dystrophy and the collagen VI disorders. Each of the X-linked form of Emery-Dreifuss muscular dystrophy and Ullrich muscular dystrophy has a prevalence of 0.13/100,000, making both very rare. Bethlem myopathy was relatively more common with a prevalence of 0.77/100,000. Overall our study provides comprehensive epidemiological information on individually rare inherited neuromuscular conditions in Northern England. Despite the deliberate exclusion of relatively common groups such as hereditary motor and sensory neuropathy (40/100,000) and mitochondrial disorders (9.2/100,000), the combined prevalence is 37.0/100,000, demonstrating that these disorders, taken as a group, encompass a significant proportion of patients with chronic disease. The study also illustrates the immense diagnostic progress since the first regional survey over 50 years ago by Walton and Nattrass.

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Year:  2009        PMID: 19767415      PMCID: PMC4038491          DOI: 10.1093/brain/awp236

Source DB:  PubMed          Journal:  Brain        ISSN: 0006-8950            Impact factor:   13.501


  43 in total

1.  Report of the 115th ENMC workshop: DM2/PROMM and other myotonic dystrophies. 3rd Workshop, 14-16 February 2003, Naarden, The Netherlands.

Authors:  B Udd; G Meola; R Krahe; C Thornton; L Ranum; J Day; G Bassez; K Ricker
Journal:  Neuromuscul Disord       Date:  2003-09       Impact factor: 4.296

2.  Anatomy of a founder effect: myotonic dystrophy in Northeastern Quebec.

Authors:  Vania Yotova; Damian Labuda; Ewa Zietkiewicz; Dominik Gehl; Alan Lovell; Jean-François Lefebvre; Stéphane Bourgeois; Emilie Lemieux-Blanchard; Marcin Labuda; Hélène Vézina; Louis Houde; Marc Tremblay; Bruno Toupance; Evelyne Heyer; Thomas J Hudson; Claude Laberge
Journal:  Hum Genet       Date:  2005-05-10       Impact factor: 4.132

3.  On the classification, natural history and treatment of the myopathies.

Authors:  J N WALTON; F J NATTRASS
Journal:  Brain       Date:  1954       Impact factor: 13.501

Review 4.  Molecular biology of distal muscular dystrophies--sarcomeric proteins on top.

Authors:  Bjarne Udd
Journal:  Biochim Biophys Acta       Date:  2006-08-18

5.  LGMD2A: genotype-phenotype correlations based on a large mutational survey on the calpain 3 gene.

Authors:  A Sáenz; F Leturcq; A M Cobo; J J Poza; X Ferrer; D Otaegui; P Camaño; M Urtasun; J Vílchez; E Gutiérrez-Rivas; J Emparanza; L Merlini; C Paisán; M Goicoechea; L Blázquez; B Eymard; H Lochmuller; M Walter; C Bonnemann; D Figarella-Branger; J C Kaplan; J A Urtizberea; J F Martí-Massó; A López de Munain
Journal:  Brain       Date:  2005-02-02       Impact factor: 13.501

6.  Clinical, molecular, and protein correlations in a large sample of genetically diagnosed Italian limb girdle muscular dystrophy patients.

Authors:  Michela Guglieri; Francesca Magri; Maria Grazia D'Angelo; Alessandro Prelle; Lucia Morandi; Carmelo Rodolico; Rachele Cagliani; Marina Mora; Francesco Fortunato; Andreina Bordoni; Roberto Del Bo; Serena Ghezzi; Serena Pagliarani; Sabrina Lucchiari; Sabrina Salani; Chiara Zecca; Costanza Lamperti; Dario Ronchi; Mohammed Aguennouz; Patrizia Ciscato; Claudia Di Blasi; Alessandra Ruggieri; Isabella Moroni; Anna Turconi; Antonio Toscano; Maurizio Moggio; Nereo Bresolin; Giacomo P Comi
Journal:  Hum Mutat       Date:  2008-02       Impact factor: 4.878

7.  Myasthenia gravis: a population based epidemiological study in Cambridgeshire, England.

Authors:  N P Robertson; J Deans; D A Compston
Journal:  J Neurol Neurosurg Psychiatry       Date:  1998-10       Impact factor: 10.154

Review 8.  Muscular dystrophies due to glycosylation defects.

Authors:  Francesco Muntoni; Silvia Torelli; Martin Brockington
Journal:  Neurotherapeutics       Date:  2008-10       Impact factor: 7.620

9.  Calpain-3 mutations in Turkey.

Authors:  Burcu Balci; Stefania Aurino; Göknur Haliloglu; Beril Talim; Sevim Erdem; Zuhal Akcören; Ersin Tan; Melda Caglar; Isabelle Richard; Vincenzo Nigro; Haluk Topaloglu; Pervin Dincer
Journal:  Eur J Pediatr       Date:  2006-01-13       Impact factor: 3.183

10.  Prevalence of mitochondrial DNA disease in adults.

Authors:  Andrew M Schaefer; Robert McFarland; Emma L Blakely; Langping He; Roger G Whittaker; Robert W Taylor; Patrick F Chinnery; Douglass M Turnbull
Journal:  Ann Neurol       Date:  2008-01       Impact factor: 10.422

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  152 in total

1.  Muscle weakness in myotonic dystrophy associated with misregulated splicing and altered gating of Ca(V)1.1 calcium channel.

Authors:  Zhen Zhi Tang; Viktor Yarotskyy; Lan Wei; Krzysztof Sobczak; Masayuki Nakamori; Katy Eichinger; Richard T Moxley; Robert T Dirksen; Charles A Thornton
Journal:  Hum Mol Genet       Date:  2011-12-02       Impact factor: 6.150

2.  Clinical utility gene card for: Central core disease.

Authors:  Suzanne Lillis; Stephen Abbs; Clemens R Mueller; Francesco Muntoni; Heinz Jungbluth
Journal:  Eur J Hum Genet       Date:  2011-10-12       Impact factor: 4.246

3.  A national profile of health care and family impacts of children with muscular dystrophy and special health care needs in the United States.

Authors:  Lijing Ouyang; Scott D Grosse; Michael H Fox; Julie Bolen
Journal:  J Child Neurol       Date:  2011-09-27       Impact factor: 1.987

Review 4.  Quality improvement in neurology: muscular dystrophy quality measures.

Authors:  Pushpa Narayanaswami; Richard Dubinsky; David Wang; Gina Gjorvad; William David; Jonathan Finder; Benn Smith; Jianguo Cheng; Frederic Shapiro; Michelle Mellion; Christopher Spurney; Jodi Wolff; John England
Journal:  Neurology       Date:  2015-09-08       Impact factor: 9.910

5.  Factors affecting the health-related quality of life of caregivers of patients with muscular dystrophy.

Authors:  Yi-Jing Lue; Shun-Sheng Chen; Yen-Mou Lu
Journal:  J Neurol       Date:  2018-04-26       Impact factor: 4.849

Review 6.  The collagen VI-related myopathies Ullrich congenital muscular dystrophy and Bethlem myopathy.

Authors:  Carsten G Bönnemann
Journal:  Handb Clin Neurol       Date:  2011

7.  A mouse model for dominant collagen VI disorders: heterozygous deletion of Col6a3 Exon 16.

Authors:  Te-Cheng Pan; Rui-Zhu Zhang; Machiko Arita; Sasha Bogdanovich; Sheila M Adams; Sudheer Kumar Gara; Raimund Wagener; Tejvior S Khurana; David E Birk; Mon-Li Chu
Journal:  J Biol Chem       Date:  2014-02-22       Impact factor: 5.157

8.  Cost Effectiveness of Nusinersen in the Treatment of Patients with Infantile-Onset and Later-Onset Spinal Muscular Atrophy in Sweden.

Authors:  Santiago Zuluaga-Sanchez; Megan Teynor; Christopher Knight; Robin Thompson; Thomas Lundqvist; Mats Ekelund; Annabelle Forsmark; Adrian D Vickers; Andrew Lloyd
Journal:  Pharmacoeconomics       Date:  2019-06       Impact factor: 4.981

9.  A recurrent COL6A1 pseudoexon insertion causes muscular dystrophy and is effectively targeted by splice-correction therapies.

Authors:  Véronique Bolduc; A Reghan Foley; Herimela Solomon-Degefa; Apurva Sarathy; Sandra Donkervoort; Ying Hu; Grace S Chen; Katherine Sizov; Matthew Nalls; Haiyan Zhou; Sara Aguti; Beryl B Cummings; Monkol Lek; Taru Tukiainen; Jamie L Marshall; Oded Regev; Dina Marek-Yagel; Anna Sarkozy; Russell J Butterfield; Cristina Jou; Cecilia Jimenez-Mallebrera; Yan Li; Corine Gartioux; Kamel Mamchaoui; Valérie Allamand; Francesca Gualandi; Alessandra Ferlini; Eric Hanssen; Steve D Wilton; Shireen R Lamandé; Daniel G MacArthur; Raimund Wagener; Francesco Muntoni; Carsten G Bönnemann
Journal:  JCI Insight       Date:  2019-03-21

10.  Keloids, Spontaneous or After Minor Skin Injury: Importance of Not Missing Bethlem Myopathy.

Authors:  Constanza Echeverría; Alejandra Diaz; Bernardita Suarez; Jorge A Bevilacqua; Carsten Bonnemann; Enrico Bertini; Claudia Castiglioni
Journal:  Acta Derm Venereol       Date:  2017-02-08       Impact factor: 4.437

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