Abstract

Introduction: Hepatic Graft-versus-Host Disease (GVHD) presenting with xanthomatous (foamy) histiocytes on cutaneous biopsy is a rare phenomenon described primarily in hematopoietic stem cell transplantation and results in high morbidity and mortality. Case Presentation: A 54-year-old male with a history of alcohol-related liver disease presented with two days of a pruritic, non-painful rash on his back, one month after multivisceral transplant (liver, pancreas, stomach, and bowel). He also noted increased ileostomy output. Physical examination found erythematous macules and papules coalescing into plaques (morbilliform) on the back. Laboratory examination was notably negative for CMV, EBV, and HCV. Liver function tests, cholesterol, and triglycerides were within normal limits. Endoscopic biopsy of the ileostomy showed no acute cellular rejection. A punch biopsy of the back was performed. Histopathology demonstrated lymphocytic interface dermatitis with rare necrotic keratinocytes. In addition, foamy histiocytic cells were noted in the superficial dermis. Short Tandem Repeat analysis demonstrated the presence of a mixed chimera consisting of 17% donor and 83% recipient DNA. Overall, these findings were consistent with GVHD confined to the skin (<25% BSA). He was treated with solumedrol for GVHD and triamcinolone ointment for symptomatic relief of the rash. Unfortunately, the patient developed severe neutropenia and died from septic shock secondary to ileostomy leak. Conclusion: We present a case of multivisceral transplant complicated by cutaneous GVHD with histologic evidence of xanthomatous histiocytes and without systemic signs of hepatic GVHD. These findings expand the spectrum of histologic changes that can occur in GVHD. Interestingly, our patient did not have hypercholesterolemia or hypertriglyceridemia in contrast to similar published cases.

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