ArticleEuropean journal of neurology2025
Split Hand Syndrome in Charcot-Marie-Tooth Disease Type X1 (CMTX1): A Clinical, Neurophysiological, and Radiological Study.
Article in European journal of neurology, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 3 papers.
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3 citing papers in PubMed.
- Beyond ALS: split-hand syndrome in immune-mediated motor neuropathies.Journal of neurology · 2026Article
- Hirayama disease: an uncommon cause of motor neuron disease.Arquivos de neuro-psiquiatria · 2026Article
- Cleft Hand and Foot Syndrome: A Report of Three Cases with Review of Literature.Journal of orthopaedic case reports · 2025Article
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11 authors.
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Abstract
backgroundSplit hand syndrome (SHS) is a hand atrophy pattern characterized by predominant wasting in the thenar muscles (abductor pollicis brevis-APB, first dorsal interosseous-FDI), with relative sparing of the hypothenar (abductor digiti minimi-ADM). SHS was also reported in CMTX1, but eventually attributed to median-ulnar dissociated involvement. We investigated the presence and specificity of SHS in CMTX1.
methodsWe gathered clinical/neurophysiological/radiological information in CMTX1 and non-CMTX1 patients, including disease severity (CMT Examination Score-CMTES) and compound muscle amplitude potential (CMAP) of APB/FDI/ADM. We obtained ADM/APB ratio (ADM/APBr) = CMAP
resultsWe evaluated 22 CMTX1 (age 41.3 ± 12.2) and 40 non-CMTX1 (49.2 ± 14.9). 50% (vs. 3% of non-CMTX1, p < 0.001) and 64% (vs. 0%, p < 0.001) of CMTX1 had clinical and neurophysiological SHS, respectively. In CMTX1, SHS was independent from gender and hand dominance. Both ADM/APBr and SHI correlated with disease duration (rs = 0.77, p < 0.001; rs = -0.65, p = 0.022, respectively), and CMTES (rs = 0.56, p = 0.006; rs = -0.62, p < 0.001, respectively). 3/8 CMTX1 had radiological SHS. Within the Italian CMT Registry cohort, ADM/APBr > 1.7 yielded a specificity of 74% for CMTX1 females among I-CMT/CMT2, and 85% for CMTX1 males among CMT1/I-CMT. DISCUSSION: Up to 59% of CMTX1 patients develop thenar-hypothenar (rather than median-ulnar) SHS. MRI is a novel approach to detect this dissociated denervation pattern. ADM/APBr > 1.7 may be useful to address GJB1 testing in males.
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