Literature DB >> 29770363

The multifaceted clinical presentation of VCP-proteinopathy in a Greek family.

George K Papadimas1, George P Paraskevas1, Thomas Zambelis1, Chrisostomos Karagiaouris1, Mara Bourbouli1, Anastasia Bougea1, Maggie C Walter2, Nicolas U Schumacher2, Sabine Krause2, Elisabeth Kapaki1.   

Abstract

VCP-proteinopathy is a multisystem neurodegenerative disorder caused by mutations in valosin containing protein. Here, we report the first Greek case of VCP-proteinopathy in a 62 year old patient with a slowly progressing muscular weakness since his mid-40s and a severe deterioration during the last year. He also manifested dementia with prominent neuropsychiatric symptoms, including aggression, apathy, palilalia and obsessions. Brain MRI revealed frontal atrophy, while muscle MRI showed diffuse muscle atrophy. Family history was positive and several members of the family had been diagnosed with motor neuron disease, dementia or behavioral symptoms. Sequencing of the VCP gene revealed a pathogenic heterozygous missense mutation p.R159H. Conclusively, the present report highlights the intrafamilial variability and broadens the phenotypic spectrum of VCP-proteinopathy.

Entities:  

Keywords:  ALS; VCP; dementia

Mesh:

Substances:

Year:  2017        PMID: 29770363      PMCID: PMC5953233     

Source DB:  PubMed          Journal:  Acta Myol        ISSN: 1128-2460


  12 in total

1.  VCP mutations in familial and sporadic amyotrophic lateral sclerosis.

Authors:  Max Koppers; Marka M van Blitterswijk; Lotte Vlam; Paulina A Rowicka; Paul W J van Vught; Ewout J N Groen; Wim G M Spliet; JooYeon Engelen-Lee; Helenius J Schelhaas; Marianne de Visser; Anneke J van der Kooi; W-Ludo van der Pol; R Jeroen Pasterkamp; Jan H Veldink; Leonard H van den Berg
Journal:  Neurobiol Aging       Date:  2011-11-10       Impact factor: 4.673

Review 2.  New ALS-Related Genes Expand the Spectrum Paradigm of Amyotrophic Lateral Sclerosis.

Authors:  Mario Sabatelli; Giuseppe Marangi; Amelia Conte; Giorgio Tasca; Marcella Zollino; Serena Lattante
Journal:  Brain Pathol       Date:  2016-03       Impact factor: 6.508

3.  Clinical delineation and localization to chromosome 9p13.3-p12 of a unique dominant disorder in four families: hereditary inclusion body myopathy, Paget disease of bone, and frontotemporal dementia.

Authors:  M J Kovach; B Waggoner; S M Leal; D Gelber; R Khardori; M A Levenstien; C A Shanks; G Gregg; M T Al-Lozi; T Miller; W Rakowicz; G Lopate; J Florence; G Glosser; Z Simmons; J C Morris; M P Whyte; A Pestronk; V E Kimonis
Journal:  Mol Genet Metab       Date:  2001-12       Impact factor: 4.797

4.  Characterization of the Asian myopathy patients with VCP mutations.

Authors:  Z Shi; Y K Hayashi; S Mitsuhashi; K Goto; D Kaneda; Y-C Choi; C Toyoda; S Hieda; T Kamiyama; H Sato; M Wada; S Noguchi; I Nonaka; I Nishino
Journal:  Eur J Neurol       Date:  2011-10-31       Impact factor: 6.089

5.  Clinical heterogeneity in 3 unrelated families linked to VCP p.Arg159His.

Authors:  J van der Zee; D Pirici; T Van Langenhove; S Engelborghs; R Vandenberghe; M Hoffmann; G Pusswald; M Van den Broeck; K Peeters; M Mattheijssens; J-J Martin; P P De Deyn; M Cruts; D Haubenberger; S Kumar-Singh; A Zimprich; C Van Broeckhoven
Journal:  Neurology       Date:  2009-08-25       Impact factor: 9.910

6.  Clinical outcome in 19 French and Spanish patients with valosin-containing protein myopathy associated with Paget's disease of bone and frontotemporal dementia.

Authors:  Tanya Stojkovic; El Hadi Hammouda; Pascale Richard; Adolfo López de Munain; Javier Ruiz-Martinez; Pilar Camaño; Pilar Camaño Gonzalez; Pascal Laforêt; Isabelle Pénisson-Besnier; Xavier Ferrer; Arnaud Lacour; Lucette Lacomblez; Kristl G Claeys; Claude-Alain Maurage; Michel Fardeau; Bruno Eymard
Journal:  Neuromuscul Disord       Date:  2009-04-11       Impact factor: 4.296

7.  215th ENMC International Workshop VCP-related multi-system proteinopathy (IBMPFD) 13-15 November 2015, Heemskerk, The Netherlands.

Authors:  Teresinha Evangelista; Conrad C Weihl; Virginia Kimonis; Hanns Lochmüller
Journal:  Neuromuscul Disord       Date:  2016-05-30       Impact factor: 4.296

8.  Clinical and molecular studies in a unique family with autosomal dominant limb-girdle muscular dystrophy and Paget disease of bone.

Authors:  V E Kimonis; M J Kovach; B Waggoner; S Leal; A Salam; L Rimer; K Davis; R Khardori; D Gelber
Journal:  Genet Med       Date:  2000 Jul-Aug       Impact factor: 8.822

Review 9.  Insights into muscle degeneration from heritable inclusion body myopathies.

Authors:  Sabine Krause
Journal:  Front Aging Neurosci       Date:  2015-02-12       Impact factor: 5.750

10.  Mutational spectrum and phenotypic variability of VCP-related neurological disease in the UK.

Authors:  S Figueroa-Bonaparte; J Hudson; R Barresi; T Polvikoski; T Williams; A Töpf; E Harris; D Hilton-Jones; R Petty; T A Willis; C Longman; C F Dougan; M J Parton; M G Hanna; R Quinlivan; M E Farrugia; M Guglieri; K Bushby; V Straub; H Lochmüller; T Evangelista
Journal:  J Neurol Neurosurg Psychiatry       Date:  2015-06-23       Impact factor: 10.154

View more
  4 in total

Review 1.  Multisystem Proteinopathy Due to VCP Mutations: A Review of Clinical Heterogeneity and Genetic Diagnosis.

Authors:  Gerald Pfeffer; Grace Lee; Carly S Pontifex; Roberto D Fanganiello; Allison Peck; Conrad C Weihl; Virginia Kimonis
Journal:  Genes (Basel)       Date:  2022-05-27       Impact factor: 4.141

2.  Phenotype of VCP Mutations in Chinese Amyotrophic Lateral Sclerosis Patients.

Authors:  Shu-Yan Feng; Han Lin; Chun-Hui Che; Hua-Pin Huang; Chang-Yun Liu; Zhang-Yu Zou
Journal:  Front Neurol       Date:  2022-02-07       Impact factor: 4.003

3.  A Novel Mutation (D395A) in Valosin-Containing Protein Gene Is Associated With Early Onset Frontotemporal Dementia in an Italian Family.

Authors:  Francesco Bruno; Maria Elena Conidi; Gianfranco Puccio; Francesca Frangipane; Valentina Laganà; Livia Bernardi; Nicoletta Smirne; Maria Mirabelli; Rosanna Colao; Sabrina Curcio; Raffaele Di Lorenzo; Raffaele Maletta; Amalia Cecilia Bruni
Journal:  Front Genet       Date:  2021-11-30       Impact factor: 4.599

Review 4.  Development of a standard of care for patients with valosin-containing protein associated multisystem proteinopathy.

Authors:  Manisha Korb; Allison Peck; Lindsay N Alfano; Kenneth I Berger; Meredith K James; Nupur Ghoshal; Elise Healzer; Claire Henchcliffe; Shaida Khan; Pradeep P A Mammen; Sujata Patel; Gerald Pfeffer; Stuart H Ralston; Bhaskar Roy; William W Seeley; Andrea Swenson; Tahseen Mozaffar; Conrad Weihl; Virginia Kimonis
Journal:  Orphanet J Rare Dis       Date:  2022-01-29       Impact factor: 4.123

  4 in total

北京卡尤迪生物科技股份有限公司 © 2022-2023.