D-Penicillamine-Induced Stevens-Johnson Syndrome in a Patient with Gold Cyanide Intoxication: A Case Report

Abstract

D-penicillamine is used as the mainstay of chelation therapy for Wilson's disease and for heavy metal intoxication. D-penicillamine itself has been noted to cause several systemic side effects as well as symptoms related to the skin. Common cutaneous side effects such as acute hypersensitivity reactions, elastic fiber abnormalities, and bullous diseases have been occasionally described. Herein, we report a case of a 23-year-old Thai female with gold intoxication who developed Stevens-Johnson syndrome (SJS) following the treatment of D-penicillamine. To our knowledge, D-penicillamine-induced SJS is exceptionally rare. To raise awareness of potentially fatal cutaneous adverse drug reaction triggered by D-penicillamine, published literature regarding SJS induced by this agent has also been reviewed. D-penicillamine should be regarded as a possible culprit in patients presenting with SJS following D-penicillamine administration and should be promptly discontinued.

Keywords: chelating agent; cutaneous adverse drug reaction; cyanide; epidermal necrolysis; gold salt; penicillamine.

Figure 1

Cutaneous manifestation of D-penicillamine-induced Stevens-Johnson syndrome. Clinical features show multiple erythematous to dusky red macules coalescing into patches on the chest, abdomen (A), and back (B) with positive Nikolsky’s sign (B, inset).

Figure 2

Multiple erythematous to dusky red macules coalescing into patches on the palms (A) and soles (B).

Figure 3

Facial involvement together with bilateral conjunctival injections (A). Multiple erosions in the oral cavity and lips, some with overlying hemorrhagic crust (B).