Pacinian corpuscles' (PCs) histological structures were first described by Paccini [1] in 1835. They are classically described as rapidly adapting mechanoreceptors, responding to the onset or cessation of mechanical stimuli. [2] Histologically, PCs are multilamellar structures with a characteristic onion-like appearance. Pathological hyperplasia of PCs is immensely rare. Pacinian corpuscle hyperplasia was first published as a Pacinian neurofibroma in 1952. [3] It is a painful disorder that mostly involves the hand. The pathogenesis of PC hyperplasia is still unclear and has not yet been defined. However, trauma may play an important role in the etiology of PC hyperplasia since a history of local trauma has been reported in 57% of the cases [4] In this case report, we present an interesting case with PC hyperplasia located on the Pacinian disorders are exceedingly rare, and the exact pathogenesis is still unknown. The most common symptoms are pain, sensory changes, and a visible or palpable mass, and diagnosis is usually made by pathological examination after the excision of the painful nodule. In this case report, we present the case of a 49-year-old male with Pacinian corpuscle hyperplasia located on the metacarpophalangeal joint, emerging at the same hand of the patient two years after the treatment due to complex regional pain syndrome (CRPS). To the best of our knowledge, this is the first case report revealing the association of CRPS with hyperplasia of Pacinian corpuscles.