[Phenylbutazone-induced sialadenitis fever simulating angioedema]

Abstract

Background: Drug-induced sialadenitis is uncommon and unrecognized. Drugs such as nitrofurantoïn, nifedipine and methimazole have been reported to induce sialadenitis. However, phenylbutazone and oxyphenbutazone are the most frequently implicated agents. We describe a case of phenylbutazone-induced parotitis and submaxillitis with cutaneous and hepatic involvement.

Case report: A 51 year-old woman who had received phenylbutazone for the past 6 days was hospitalized for diagnosis of Quincke's oedema. Clinical examination in fact revealed bilateral parotitis and submaxillitis. The patient had contracted mumps in infancy. Improvement was noticed 8 days after stopping the drug and treatment by glucocorticosteroid. Nevertheless a pruritic eruption with fever appeared. Laboratory data showed leukocytosis with neutrophilia, ESR of 75 mm/hr, hepatic cholestasis and cytolysis. Infectious and autoimmune causes were ruled out. The eruption spontaneously disappeared after 5 days. Laboratory studies 3 weeks later were normal.

Discussion: Quincke's edema diagnosis had been established too fast on "allergic past history" and patient interrogation. Complete clinical examination revealed the correct diagnosis of sialadenitis. This observation shows similarities with other publications: unbearable xerostomia appearing before sialadenitis and with a long course, parotitis with sub-maxillitis, 6 days delay after the first administration of phenylbutazone before fever, local evolution without complication, inflammatory biological syndrome with neutrophilia and absence of infectious cause. Pruritic maculo-papulous eruption and biological hepatic abnormalities are however rare. An hypersensibility mechanism is discussed.