Biallelic mutations in the SBF1 gene have been identified in one family with demyelinating Charcot-Marie-Tooth disease (CMT4B3) and two families with axonal neuropathy and additional neurological and skeletal features. Here we describe novel sequence variants in SBF1 (c.1168C>G and c.2209_2210del) as the potential causative mutations in two siblings with severe axonal neuropathy, hearing loss, facial weakness and bulbar features. Pathogenicity of these variants is supported by co-segregation and in silico analyses and evolutionary conservation. Our findings suggest that SBF1 mutations may cause a syndromic form of autosomal recessive axonal neuropathy (AR-CMT2) in addition to CMT4B3.
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http://dx.doi.org/10.1007/s10048-016-0505-1 | DOI Listing |
Cureus
December 2024
Internal Medicine, Queen's Hospital, Barking, Havering and Redbridge University Hospitals NHS Trust, Romford, GBR.
Diabetic lumbosacral radiculoplexus neuropathy, also known as diabetic lumbosacral plexopathy or diabetic amyotrophy, is a rare complication of diabetes mellitus. Due to its varied clinical presentation and wide differential, it may pose a diagnostic quandary in assessing patients with proximal asymmetrical lower limb weakness. We present the case of a 74-year-old female patient with a recent onset of falls and aim to discuss the aetiology, differentials, and treatment modalities in diabetic plexopathy.
View Article and Find Full Text PDFClin Ther
December 2024
Neurology Department, The Walton Centre NHS Foundation Trust, Liverpool, United Kingdom; Institute of Systems, Molecular and Integrative Biology, University of Liverpool, Liverpool, United Kingdom; Department of Psychology, Manchester Metropolitan University, Manchester, United Kingdom.
Purpose: An increased prevalence of peripheral polyneuropathy (PN) in Parkinson's disease (PD) associated with greater functional impairment has previously been reported. A possible cause has been suggested as levodopa therapy. The aim of this real-world study was to assess the prevalence and the characteristics of PN in PD and to investigate the putative association between PN and oral levodopa.
View Article and Find Full Text PDFClin Neurophysiol
December 2024
Department of Clinical Neurophysiology, Vall d'Hebron University Hospital, Passeig de la Vall d'Hebron, 119, 08035 Barcelona, Spain. Electronic address:
Introduction/objective: Biallelic expansion of the pentanucleotide AAGGG in the RFC1- gene is associated with cerebellar ataxia, neuropathy, and vestibular areflexia syndrome (CANVAS). This study aimed to comprehensively characterise this condition by conducting an in-depth neurophysiological examination of afflicted patients.
Methods: A retrospective analysis was conducted in 31 RFC1-positive patients.
eNeurologicalSci
March 2025
Department of Neurology, University of Michigan, Ann Arbor, MI 48109, USA.
Obesity and the metabolic syndrome (MetS) are major global health challenges that contribute significantly to the rising prevalence of type 2 diabetes (T2D) and neuropathy. Neuropathy, a common and disabling complication of T2D, is characterized by progressive distal-to-proximal axonal degeneration, driven in part by mitochondrial dysfunction in both neurons and axons. Recent evidence points to the toxic effects of saturated fatty acids on peripheral nerve health, with studies demonstrating that these fats impair mitochondrial function and bioenergetics, leading to distal axonal loss.
View Article and Find Full Text PDFJ Pharmacol Sci
January 2025
Department of Clinical Pharmacy and Pharmaceutical Care, Graduate School of Pharmaceutical Sciences, Kyushu University, Fukuoka, 812-8582, Japan.
Paclitaxel induces peripheral neuropathy, which is considered a dose-limiting factor. However, appropriate prophylactic agents are currently unavailable. We investigated the prophylactic effects of calmangafodipir, a superoxide dismutase mimetic, on paclitaxel-induced peripheral neuropathy using a male rat model.
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