Literature DB >> 28680318

Clinical, Electrophysiological, and Serological Evaluation of Patients with Cramp-Fasciculation Syndrome.

Mürüvvet Poyraz1, Zeliha Matur2, Fikret Aysal1, Erdem Tüzün3, Lütfü Hanoğlu1, A Emre Öge3.   

Abstract

INTRODUCTION: Cramp-fasciculation syndrome (CFS) is a rare peripheral nerve hyperexcitability syndrome. There are only a few reports on clinical and serological profile of a CFS cohort that was followed up by a single outpatient clinic.
METHODS: Clinical, electrophysiological, and serological features of 6 CFS patients (5 men, 1 woman; 27-65 years old) were investigated.
RESULTS: All patients presented with cramps, fasciculations, muscle pain, and autonomic symptoms, and 2 also reported numbness and burning sensation in limbs, suggestive of neuropathic pain. Antibodies to uncharacterized voltage-gated potassium channel (VGKC)-complex proteins were found in 2 patients and to contactin-associated protein-like 2 (CASPR2) in 1 patient. None of the patients had a tumor. Most of the patients revealed prolonged after-discharges following tibial nerve stimulation. Nerve conduction studies and R-R interval variability tests were normal, whereas sympathetic skin responses were increased in amplitude in 3 seronegative patients. Five patients showed favorable response to carbamazepine or pregabalin treatment, whereas 1 VGKC-antibody-positive patient was resistant to carbamazepine and immunosuppressant treatment.
CONCLUSION: Neuropathic pain and VGKC-complex antibodies may be encountered in CFS patients. Although autonomic symptoms are commonly found in CFS, routine autonomic system tests which are done in electrophysiology laboratories might yield normal results.

Entities:  

Keywords:  Cramp-fasciculation syndrome (CFS); neuropathic pain; peripheral nerve hyperexcitability; voltage-gated potassium channel (VGKC)-complex proteins

Year:  2016        PMID: 28680318      PMCID: PMC5491670          DOI: 10.5152/npa.2016.14816

Source DB:  PubMed          Journal:  Noro Psikiyatr Ars        ISSN: 1300-0667            Impact factor:   1.339


  10 in total

1.  Sympathetic skin response: normal results in different experimental conditions.

Authors:  B Elie; P Guiheneuc
Journal:  Electroencephalogr Clin Neurophysiol       Date:  1990-09

2.  Cramp-fasciculation syndrome: a treatable hyperexcitable peripheral nerve disorder.

Authors:  A J Tahmoush; R J Alonso; G P Tahmoush; T D Heiman-Patterson
Journal:  Neurology       Date:  1991-07       Impact factor: 9.910

3.  Cramp-fasciculation syndrome in patients with and without neural autoantibodies.

Authors:  Teerin Liewluck; Christopher J Klein; Lyell K Jones
Journal:  Muscle Nerve       Date:  2013-12-17       Impact factor: 3.217

4.  Bronchial involvement in the cramp-fasciculation syndrome.

Authors:  Manuel de Entrambasaguas; Juan-José Ortega-Albás; María-Dolores Martínez-Lozano; José-Ramón Díaz
Journal:  Eur Neurol       Date:  2006-09-08       Impact factor: 1.710

5.  RR interval variation and the sympathetic skin response in the assessment of autonomic function in peripheral neuropathy.

Authors:  B T Shahani; T J Day; D Cros; N Khalil; C S Kneebone
Journal:  Arch Neurol       Date:  1990-06

Review 6.  Peripheral nerve hyperexcitability syndromes.

Authors:  Cem Ismail Küçükali; Murat Kürtüncü; Halil İbrahim Akçay; Erdem Tüzün; Ali Emre Öge
Journal:  Rev Neurosci       Date:  2015       Impact factor: 4.353

Review 7.  Autoimmune disorders of neuronal potassium channels.

Authors:  John Newsom-Davis; Camilla Buckley; Linda Clover; Ian Hart; Paul Maddison; Erdem Tüzüm; Angela Vincent
Journal:  Ann N Y Acad Sci       Date:  2003-09       Impact factor: 5.691

8.  Phenotypic variants of autoimmune peripheral nerve hyperexcitability.

Authors:  Ian K Hart; Paul Maddison; John Newsom-Davis; Angela Vincent; Kerry R Mills
Journal:  Brain       Date:  2002-08       Impact factor: 13.501

9.  The sympathetic skin response: normal values, elucidation of afferent components and application limits.

Authors:  A Uncini; S L Pullman; R E Lovelace; D Gambi
Journal:  J Neurol Sci       Date:  1988-11       Impact factor: 3.181

10.  Antibodies to Kv1 potassium channel-complex proteins leucine-rich, glioma inactivated 1 protein and contactin-associated protein-2 in limbic encephalitis, Morvan's syndrome and acquired neuromyotonia.

Authors:  Sarosh R Irani; Sian Alexander; Patrick Waters; Kleopas A Kleopa; Philippa Pettingill; Luigi Zuliani; Elior Peles; Camilla Buckley; Bethan Lang; Angela Vincent
Journal:  Brain       Date:  2010-07-27       Impact factor: 13.501

  10 in total
  2 in total

Review 1.  Current Treatment Options for Peripheral Nerve Hyperexcitability Syndromes.

Authors:  Cheran Elangovan; Adeolu Morawo; Aiesha Ahmed
Journal:  Curr Treat Options Neurol       Date:  2018-05-28       Impact factor: 3.972

2.  Genetic rhabdomyolysis within the spectrum of the Spinocerebellar Ataxia type 2 responsive to pregabalin.

Authors:  Fabian Rossi; Joe Ma; Nina Tsakadze; Lourdes Benes-Lima; Julio Araque Gonzalez; Michael Hoffmann
Journal:  Cerebellum Ataxias       Date:  2021-03-05
  2 in total

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