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Case Reports

Abstract

A 59-year-old woman with Stage IV accelerated chronic lymphocytic leukemia (CLL) on rituximab, IVIG, filgrastim, and venetoclax presented with an acute morbilliform eruption progressing to erythroderma after prolonged hospitalization for septic shock and respiratory failure. During the preceding three weeks, she had received numerous medications including IV vancomycin, ceftriaxone, furosemide, meropenem, piperacillin-tazobactam, iodinated contrast, and posaconazole followed by isavuconazole. She reported five days of cephalocaudal spread of pruritic, burning and erythematous rash, along with oral ulcers, conjunctival discomfort, and dysuria. Pre-existing DSAP lesions on the distal extremities concurrently became purpuric. No lymphadenopathy, bullae, or necrosis were observed. Vitals showed tachycardia. Laboratories demonstrated pancytopenia, with eosinophils within reference limits. Microbiology serologies and cultures were negative. Histopathology revealed a superficial perivascular and intravascular atypical T-cell infiltrate with purpura, pseudovesiculation, eosinophils, and sparse foamy histiocytes, suggestive of an atypical cutaneous T-cell leukemia/lymphoma. Ki-67 highlighted T cells and was focally near 90%, while CD20, PAX5, TdT, CD21, CD56, and CD138 were negative. Tissue, blood, and marrow studies were negative for T-cell receptor gamma gene rearrangements. IV methylprednisolone was started upon admission. After resolution of the rash over the following week, repeat biopsy showed focal vacuolar interface dermatitis with limited residual lymphocytes. The diagnosis was concluded to represent the extreme upper limit of reactive T-cell atypia in a severe drug eruption, without a definitive culprit. The phenomenon of atypical lymphocytosis can be a dilemma in severe cutaneous adverse reactions, particularly when overt clinical criteria for DRESS are lacking in a patient with known hematologic disease.