Successful Treatment of Hydroxychloroquine-Induced Recalcitrant Acute Generalized Exanthematous Pustulosis with Cyclosporine: Case Report and Literature Review

Abstract

Acute generalized exanthematous pustulosis (AGEP) is a cutaneous reaction principally induced by drugs. Spontaneous resolution is observed in most patients. However, severe cases required systemic corticosteroid administration. Hydroxychloroquine, which is used to treat some dermatologic and rheumatologic diseases because of its anti-inflammatory and immunosuppressive effects, is an uncommon cause of AGEP. A 67-year-old female patient presented with severe AGEP due to hydroxychloroquine treatment. She was recalcitrant to supportive care and systemic corticosteroid treatment butwas successfully treated with cyclosporine. Hydroxychloroquine-induced AGEP occurs in women with underlying rheumatologic diseases, has a longer latent period, and has a severe course usually requiring systemic treatment.

A case of recalcitrant acute generalized exanthematous pustulosis with Sjogren's syndrome: Successfully treated with low‐dose cyclosporine

“…A total of twenty‐one cases of AGEP with HCQ following a PubMed search can be found in the literature . And, there are three cases reported as corticosteroid‐refractory AGEP induced by HCQ successfully treated with CsA …”

“…Corticosteroids are not always administered in most cases. CsA is not the first‐line therapy for AGEP but it was used for cases which were recalcitrant to corticosteroid therapy in the literature . Table summarizes previously reported three cases of AGEP with HCQ recalcitrant to corticosteroid therapy.…”

“…7 And, there are three cases reported as corticosteroid-refractory AGEP induced by HCQ successfully treated with CsA. [8][9][10] There are two different latency periods for AGEP in casecontrol studies, that is, 1 day for antibiotics and 11 days with a range of 4-30 days for nonantibiotics including HCQ. HCQinduced AGEP cases are generally recalcitrant to standard treatments and need a longer duration.…”

“…CsA is not the first-line therapy for AGEP but it was used for cases which were recalcitrant to corticosteroid therapy in the literature. [8][9][10] Table 1 summarizes previously reported three cases of AGEP with HCQ recalcitrant to corticosteroid therapy. In present case, we gave CsA at lower doses and in shorter periods than the reported cases.…”

Cyclosporine for corticosteroid-refractory acute generalized exanthematous pustulosis due to hydroxychloroquine

“…Table summarizes previously reported cases of corticosteroid‐refractory AGEP due to HCQ. Including the patient described herein, a history of autoimmune connective tissue disease (AICTD), latency following initial exposure of more than 15 days, and response to CsA (initiated at 4–5 mg/kg/day and tapered over several months) are features common to all three cases (Di Lernia, Grenzi, Guareschi, & Ricci, ; Yalçın, Çakmak, & Yıldırım, ).…”