The patient was a 25-year-old woman with a medical history of bilateral lung transplantation in 2012 for end-stage lung disease secondary to cystic fibrosis, chronic renal insufficiency, diabetes mellitus, exocrine pancreatic insufficiency, and chronic sinusitis. Her immunosuppressive therapy consisted of prednisolone at 10 mg/day and tacrolimus at 15.5 mg/day. Two years after lung transplantation and 2 weeks after a sinus surgery, she was admitted for fever, acute sinusitis, a nodular lesion on the hard palate, and several nodules of the inferior limbs with sporotrichoid distribution (Fig. 1A and B). Laboratory tests showed a lymphopenia. Multiple microbiological tests were performed on skin biopsies, blood, bronchoalveolar fluid, and bone marrow, using direct examination, culture, PCR, serological testing, and/or antigen detection. Numerous bacterial (tuberculosis, nontuberculous mycobacteria, syphilis, Bartonella, Coxiella, Rickettsia, Francisella, etc.), viral (HIV, viral hepatitis, cytomegalovirus, parvovirus, human herpesvirus 6, etc.), fungal (aspergillosis, cryptococcosis, hyphomycosis, etc.), and parasitic (Leishmania, Toxoplasma gondii) infections were ruled out, as the microbiological assessment results remained negative except for the observation of Pneumocystis jirovecii cysts in bronchoalveolar lavage fluid. Despite administration of broad-spectrum antibiotics for 11 days with ceftazidime at 6 g/day, ciprofloxacin at 500 mg twice a day, linezolid at 600 mg twice a day, and intravenous liposomal amphotericin B at 3 mg/kg of body weight/day, the cutaneous nodules and the oral nodule continued to spread, becoming inflammatory and necrotic; perforation of the hard palate occurred. Multiple skin biopsies were performed, revealing inflammatory reaction and necrotic tissues, with no bacterial or fungal agents. A biopsy of the lesion of the palate revealed ulcerated and necrotic tissues, with the presence of 20-µm cellular structures containing round nuclei with prominent nucleoli and vacuolated cytoplasm (Fig. 1C to F). The patient’s condition deteriorated, resulting in death 2 months after admission and 4 days after initiation of liposomal amphotericin B.
(A and B) Necrotic nodules of the inferior limbs. (C to F) Histology of the biopsy of the palate lesion revealing hemorrhagic ulcerated and necrotic tissues, with the presence of cellular structures (indicated by arrows) containing round nuclei with prominent nucleoli and vacuolated cytoplasm on hematoxylin and eosin (H&E) staining (C, E, and F) and immunohistochemical staining using antibodies against Entamoeba spp. (D). Macrophages are indicated by a square.
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For answer and discussion, see https://doi.org/10.1128/JCM.00089-18 in this issue.
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