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Servelle-Martorell syndrome is characterized by venous or rarely arterial malformations with limb hypertrophy and bony hypoplasia. Extensive involvement of the upper limb is a rare feature of Servelle-Martorell syndrome. Cases with minimal upper limb involvement have been described in the literature. A young man presented with gradually increasing multiple swellings over the right upper limb and periscapular region with functional difficulty. The arm muscles were atrophic. The bones of forearm and hand were hypoplastic and tender. We report a case of Servelle-Martorell syndrome with extensive involvement of the entire right upper limb and periscapular region.