Case report: Episodic ataxia without ataxia?

被引:1
|
作者
Gaudio, Andrea [1 ]
Gotta, Fabio [1 ]
Ponti, Clarissa [1 ,2 ]
Sanguineri, Francesca [1 ,2 ]
Trevisan, Lucia [2 ,3 ]
Geroldi, Alessandro [2 ]
Patrone, Serena [2 ]
Gemelli, Chiara [4 ]
Cabona, Corrado [5 ]
Astrea, Guja [6 ]
Fiorillo, Chiara [2 ,7 ]
Gustincich, Stefano [8 ]
Grandis, Marina [2 ,4 ]
Mandich, Paola [1 ,2 ]
机构
[1] IRCCS Osped Policlin San Martino, UOC Genet Med, Genoa, Italy
[2] Univ Genoa, Dept Neurosci Rehabil Ophthalmol Genet Maternal &, Genoa, Italy
[3] IRCCS Osped Policlin San Martino, SS Ctr Tumori Ereditari, Genoa, Italy
[4] IRCCS Osped Policlin San Martino, UOC Clin Neurol, Genoa, Italy
[5] IRCCS Osped Policlin San Martino, UOC Neurofisiopatol, Genoa, Italy
[6] IRCCS Fdn Stella Maris, Pisa, Italy
[7] IRCCS Ist Giannina Gaslini, UOC Neuropsichiatria Infantile, Genoa, Italy
[8] Ist Italiano Tecnol, Dept Neurosci & Brain Technol, Genoa, Italy
来源
FRONTIERS IN NEUROLOGY | 2023年 / 14卷
关键词
hereditary myopathies; UBR4; HSPG2; episodic ataxia; genetic modifiers; genetic testing; PERLECAN; ACETYLCHOLINESTERASE; UBR4;
D O I
10.3389/fneur.2023.1224241
中图分类号
R74 [神经病学与精神病学];
学科分类号
摘要
Hereditary myopathies represent a clinically and genetically heterogeneous group of neuromuscular disorders, characterized by highly variable clinical presentations and frequently overlapping phenotypes with other neuromuscular disorders, likely influenced by genetic and environmental modifiers. Genetic testing is often challenging due to ambiguous clinical diagnosis. Here, we present the case of a family with clinical and Electromyography (EMG) features resembling a myotonia-like disorder in which Whole Exome Sequencing (WES) analysis revealed the co-segregation of two rare missense variants in UBR4 and HSPG2, genes previously associated with episodic ataxia 8 (EA8). A review of the literature highlighted a striking overlap between the clinical and the molecular features of our family and the previously described episodic ataxias (EAs), which raises concerns about the genotype-phenotype correlation, clinical variability, and the confounding overlap in these groups of disorders. This emphasizes the importance of thoroughly framing the patient's phenotype. The more clear-cut the diagnosis, the easier the identification of a genetic determinant, and the better the prognosis and the treatment of patients.
引用
收藏
页数:8
相关论文
共 50 条
  • [1] Episodic ataxia: A case report and review of literature
    Singhvi, JP
    Prabhakar, S
    Singh, P
    NEUROLOGY INDIA, 2000, 48 (01) : 78 - 80
  • [2] Ophthalmoplegia without ataxia: a case report
    Sarilar, A. C.
    Akbostanci, M. C.
    Isikay, C. Togay
    EUROPEAN JOURNAL OF NEUROLOGY, 2012, 19 : 439 - 439
  • [3] Inherited episodic ataxia type 2 in pregnancy: A case report
    Mascherpa, Margaret
    Fichera, Anna
    Orabona, Rossana
    Recupero, Daniela
    Borroni, Barbara
    Odicino, Franco Edoardo
    Prefumo, Federico
    INTERNATIONAL JOURNAL OF GYNECOLOGY & OBSTETRICS, 2024, 165 (01) : 387 - 389
  • [4] Acute Ophthalmoplegia without Ataxia Case Report
    Bhagat, Shyam
    Oh, Seok Yoon
    Wei, Victoria
    Annesi, Thomas
    Fallon, John
    Aron, Aishi
    Rosen, Noah
    ANNALS OF NEUROLOGY, 2024, 96 : S89 - S89
  • [5] Symptomatic episodic ataxia: One case
    Anheim, M.
    Boulay, C.
    Marcel, C.
    Tranchant, C.
    MOVEMENT DISORDERS, 2008, 23 (01) : S150 - S150
  • [6] Sleep benefit in a case of Episodic ataxia
    Nagappa, Madhu
    Mundlamuri, Ravindranadh Chowdary
    Satishchandra, P.
    Pal, Pramod Kumar
    PARKINSONISM & RELATED DISORDERS, 2012, 18 (05) : 662 - 663
  • [7] Differentiating episodic ataxia type 2 from migraine: a case report
    Wu, H. J.
    Lau, W. L.
    Chan, Tina Y. C.
    Chen, Sammy P. L.
    Ko, C. H.
    HONG KONG MEDICAL JOURNAL, 2020, 26 (06) : 526 - 527
  • [8] A Case of GLUT1 Deficiency Associated Episodic Ataxia Without Epilepsy
    Tchapyjnikov, Dmitry
    Mikati, Mohamed
    NEUROLOGY, 2017, 88
  • [9] A case with episodic ataxia and multiple paroxysmal dyskinesias
    Demirkiran, M
    Yerdelen, D
    Uysal, H
    Sarica, Y
    MOVEMENT DISORDERS, 2002, 17 : S229 - S229
  • [10] Episodic ataxia type 2 - case reportll
    Matusiak-Smura, Kinga
    Kiedrzynska, Weronika
    Oborzynski, Jaroslaw
    Klimek, Andrzej
    AKTUALNOSCI NEUROLOGICZNE, 2008, 8 (02): : 106 - 108