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Invasive Oral Tongue Mucormycosis Rapidly Presenting After Orthotopic Liver Transplant

A 68-year-old male with a history of hepatitis C cirrhosis and hepatocellular carcinoma treated with transarterial chemoembolization presented in septic shock secondary to cholangitis requiring a staged orthotopic liver transplant (OLT). On the day of the first stage of his OLT, he received a dose of Basiliximab, a chimeric mouse-human monoclonal antibody against interleukin-2T cell receptors. His absolute neutrophil count also reached a nadir of 1.4 10/uL but normalized the next day. Four days later, he was given 20 mg of prednisone followed by the last stage of his transplantation. He was then started on daily tacrolimus. The very next day, before reaching therapeutic levels of tacrolimus, he was noted to have dark discoloration of his anterior oral tongue. This remained stable over the next 4 days, but given its lack of resolution, head and neck surgery was consulted to evaluate and biopsy. Figure 1 demonstrates the patients grossly necrotic oral tongue lesion. Within 24 hours, biopsy results were noted to be consistent with invasive oral tongue mucormycosis. Computed tomography sinus was clear of any sinus disease. The patient was then taken urgently for partial glossectomy and tracheostomy. Final surgical pathology, as shown in Figures 2 and 3, demonstrated necrosis and numerous fragments of broad nonseptate fungal organisms, consistent with invasive oral tongue mucormycosis. Surgical margins were clear. The patient was then placed on amphotericin B liposome and caspofungin with good response. The mucormycosis did not return, however, the patient expired 1 month later from cardiogenic versus septic shock. Mucorales fungi, of the class Zygomycetes, cause the lifethreatening fungal infection known as mucormycosis. Mononuclear and polymorphonuclear phagocytes are responsible for killing mucor subspecies in the immunocompetent host. Invasive mucormycosis increasingly affects immunocompromised patients with predisposing conditions, including organ transplantation, poorly controlled diabetes mellitus, malignancy, neutropenia, and iron overload. The estimated prevalence of mucormycosis among solid organ transplantation (SOT) recipients ranges from 0.4% to 16.0%, with incidence of 0% to 1.6% in liver transplant recipients. The mortality rate in SOT recipients with mucormycosis is high, typically ranging from 49% to 72.7%. The mortality of patients with mucormycosis infections after liver transplantation was 62.5% based on the literature review by Davari et al. The incidence of invasive mucormycosis in the United States has been estimated at about 500 cases per year. What was once a communityacquired disease, it is increasingly nosocomial in the setting of malignancy or iatrogenic immunosuppression. More alarming is the apparent implication of wooden tongue depressors and prophylactic antifungal therapy such as itraconazole or voriconazole. Figure 1. In situ grossly necrotic oral tongue. Intraoperative image depicting isolated grossly necrotic anterior oral tongue with black eschar just before wide local excision. Anterior aspect of oral tongue was excised using monopolar cautery until healthy well-perfused tongue remained posteriorly.