Pramipexole-induced limb dystonia and its associated complex regional pain syndrome in idiopathic Parkinson's disease: A case report

Abstract

Rationale: This case may be due to basal ganglia dysfunction, which was probably caused by abnormal activation of dopamine 1-like receptor (D1R) boosted by pramipexole binding on dopamine 3-like receptor (D3R) in a situation where D3R was overexpressed by the chronic treatment of L-dopa.

Patient concerns: Striatal hand and foot deformities.

Diagnoses: Striatal hand and foot deformities with CRPS.

Interventions: Steroid treatemnt and withdrawal of the pramipexole.

Outcomes: Recovered significantly.

Lessons: Since the degree of overexpression of D3R is increased in a high dose of pramipexole, for patients with PD who are treated with L-dopa chronically, a new use of pramipexole and an increase in dose to alleviate the symptoms of PD should be implemented with caution while closely observing the occurrence of drug-induced complications such as dystonia and CRPS.

Figure 1

Pramipexole-associated limb dystonia. (A) Both hands. Arrow: swelling and redness of left hands. Arrowhead: striatal hand deformity with flexion of metacarpal, proximal interphalangeal, and distal interphalageal joints. (B) Both feet. Arrow: extension of great toe with flexion of other toe.

Figure 2

(A) Three phase bone scintigraphy. Increased uptake of the left wrist and MCP joint was shown in the delayed phase. (B) Brain PET scan using F-18 FP-CIT 6.40 mCi. Decreased uptake of rostrocaudal gradient in the bilateral posterior putamen, which was consistent with idiopathic Parkinson disease. (C)(D) Diffusion brain MRI. There was no acute lesion in the diffusion brain MRI. F-18 FP-CIT = Fluorine-18 Fluorinated N-3-fluoropropyl-2-beta-carboxymethoxy-3-beta-(4-iodophenyl) nortropane, MCP = metacarpal phalangeal, MRI = magnetic resonance image, MTP = metacarpal phalangeal, PET = positron emission tomography.