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Hube, Larissa [Author]; Dohrn, Maike F. [Author]; Karsai, Gergely [Author]; Hirshman, Sarah [Author]; Van Damme, Philip [Author]; Schulz, Jörg B. [Author]; Weis, Joachim [Author]; Hornemann, Thorsten [Author]; Claeys, Kristl G. [Author]

Metabolic Syndrome, Neurotoxic 1-Deoxysphingolipids and Nervous Tissue Inflammation in Chronic Idiopathic Axonal Polyneuropathy (CIAP)

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  • Media type: E-Article
  • Title: Metabolic Syndrome, Neurotoxic 1-Deoxysphingolipids and Nervous Tissue Inflammation in Chronic Idiopathic Axonal Polyneuropathy (CIAP)
  • Contributor: Hube, Larissa [Author]; Dohrn, Maike F. [Author]; Karsai, Gergely [Author]; Hirshman, Sarah [Author]; Van Damme, Philip [Author]; Schulz, Jörg B. [Author]; Weis, Joachim [Author]; Hornemann, Thorsten [Author]; Claeys, Kristl G. [Author]
  • imprint: PLoS, 2017
  • Published in: PLoS one 12(1), e0170583 - (2017). doi:10.1371/journal.pone.0170583
  • Language: English
  • DOI: https://doi.org/10.1371/journal.pone.0170583
  • ISSN: 1932-6203
  • Origination:
  • Footnote: Diese Datenquelle enthält auch Bestandsnachweise, die nicht zu einem Volltext führen.
  • Description: AimChronic idiopathic axonal polyneuropathy (CIAP) is a slowly progressive, predominantly sensory, axonal polyneuropathy, with no aetiology being identified despite extensive investigations. We studied the potential role of the metabolic syndrome, neurotoxic 1-deoxysphingolipids (1-deoxySLs), microangiopathy and inflammation in sural nerve biopsies.MethodsWe included 30 CIAP-patients, 28 with diabetic distal symmetrical polyneuropathy (DSPN) and 31 healthy controls. We assessed standardised scales, tested for the metabolic syndrome, measured 1-deoxySLs in plasma, performed electroneurography and studied 17 sural nerve biopsies (10 CIAP; 7 DSPN).ResultsOne third of the CIAP-patients had a metabolic syndrome, significantly less frequent than DSPN-patients (89%). Although the metabolic syndrome was not significantly more prevalent in CIAP compared to healthy controls, hypercholesterolemia did occur significantly more frequent. 1-deoxySLs were significantly and equally elevated in both patient groups compared to healthy controls. Mean basal lamina thickness of small endoneurial vessels and the number of CD68- or CD8-positive cells in biopsies of CIAP- and DSPN-patients did not differ significantly. However, the number of leucocyte-common-antigen positive cells was significantly increased in CIAP.ConclusionsA non-significant trend towards a higher occurrence of the metabolic syndrome in CIAP-patients compared to healthy controls was found. 1-deoxySLs were significantly increased in plasma of CIAP-patients. Microangiopathy and an inflammatory component were present in CIAP-biopsies.
  • Access State: Open Access