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Year : 2014  |  Volume : 3  |  Issue : 2  |  Page : 114-117

Focal palmoplantar keratoderma with mutilating arthropathy: An interesting case report

Bendapudi Venkata Ramachandra, Krishna PV Rao, V Hithyshi, Lakshmi S Jhansi, India

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V Hithyshi
303, Chandrapriya Residency, Adimurthy Nagar, Anantapur - 515 001, Andhra Pradesh
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/2277-8632.134860

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Vohwinkel syndrome, characterized classically by a triad of diffuse honeycomb hyperkeratosis of palms and soles; star-shaped hyperkeratosis on the dorsa of hands, feet, knees, and elbows; and pseudoainhum is a rare type of hereditary form of palmoplantar keratoderma (PPK) which can present as two classical variants: Deafness associated variant and ichthyosis associated variant. Here we report a rare variant of hereditary PPK in a 22-year-old male patient presenting as focal PPK with striate hyperkeratosis on elbows and knees and mutilating arthropathy in addition to deafness and prognathism, the diagnosis being supported by histopathology, X-rays, and genetic mapping showing connexin defect. There are umpteen numbers of genetically inherited PPK syndromes associated with closely mimicking cutaneous features, autoamputation, and a wide range of systemic manifestations, where the differentiation of specific entities is always a difficult task. The present case is probably the first of its kind being reported from India.

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