CLINICAL CHALLENGES AND IMAGES IN GI A Rare Esophageal Complication After Autologous Stem Cell Transplantation Alexander Lemmer, Leila Kia, and Greg Cohen Division of Gastroenterology and Hepatology, Department of Medicine, Feinberg School of Medicine, Northwestern University, Chicago, Illinois
Question: A 58-year-old white man with refractory stage IIIA immunoglobulin G kappa multiple myeloma (MM) developed severe nausea, vomiting, and coffee-ground emesis during an extended hospitalization after salvage stem cell transplantation (SCT). The patient was initially diagnosed with MM 5 years before presentation, and had been treated with multiple antineoplastic therapies, including bortezomib/dexamethasone, lenalidomide, cyclophosphamide, pomalidomide, daratumumab, carﬁlzomib, and a failed autologous SCT. He subsequently underwent salvage autologous SCT with high-dose melphalan conditioning. The patient’s posttransplant course was notable for grade 4 mucositis with nausea, vomiting, and diarrhea. Infectious workup for his persistent symptoms, including testing for Clostridium difﬁcile, fecal leukocytes, Cyclospora, Giardia, Isospora, Cryptosporidium, Microsporidia, stool cultures, and ova and parasites was negative. CT of the abdomen with oral contrast was unremarkable. Supplemental enteric feeding via Dobhoff tube was initiated and symptom control was achieved with scheduled loperamide. He was discharged after adequate engraftment off immunosuppression 30 days after SCT on valacyclovir, pentamidine, micafungin, and posaconazole for prophylaxis. The patient was readmitted 1 day after discharge with severe nausea, vomiting, and coffee-ground emesis. His examination demonstrated once again severe mucositis of the oropharyngeal cavity. His laboratory results were signiﬁcant for a hemoglobin level of 7.8 g/dL and a platelet count of 13,000/mL, which were not markedly different from recent baseline values. An esophagogastroduodenoscopy was performed, which revealed 3 large, cratered esophageal ulcers with adherent exudates in the upper and middle third of the esophagus without stigmata of bleeding (Figures A, B). Biopsies of the center and the edge were taken for histologic examination (Figure C). Diffuse gastropathy and duodenopathy with a few duodenal erosions were also seen, likely contributing to upper gastrointestinal bleeding in the setting of thrombocytopenia. The etiology of esophageal ulcerations remained unclear. What is the diagnosis? Look on page 718 for the answer and see the Gastroenterology web site (www.gastrojournal.org) for more information on submitting your favorite image to Clinical Challenges and Images in GI.
Conﬂicts of interest The authors disclose no conﬂicts. © 2015 by the AGA Institute 0016-5085/$36.00 http://dx.doi.org/10.1053/j.gastro.2014.12.041
CLINICAL CHALLENGES AND IMAGES IN GI Answer to Image 4 (page 717): Esophageal Aspergillosis in an Immunocompromised Patient With Multiple Myeloma Status Post Autologous Stem Cell Transplantation Aspergillosis, or invasive Aspergillus disease, is an uncommon, but not rare, disease of immunocompromised states. Invasive aspergillosis is thought to occur by colonization of the airways, which in the setting of immunosuppression, can hematogenously disseminate to distant organs.1 The incidence of invasive aspergillosis is thought to be between 1.1% and 5.3% per year with the vast majority of cases involving the lungs or sinus cavities.1,2 Gastrointestinal presentations of disseminated aspergillosis are exceedingly rare and the majority of cases have been documented in the small bowel.1 Esophageal aspergillosis has been described in a few case reports in the literature, primarily in patients with acute myeloid leukemia undergoing treatment with chemotherapy. One case of esophageal aspergillosis has been described after allogeneic bone marrow transplantation for acute myeloid leukemia.3 To our knowledge, this is the ﬁrst case of esophageal aspergillosis described after autologous SCT. Gastrointestinal aspergillosis should be considered in immunosuppressed patients with gastrointestinal symptoms or signs of infection and ulcerations on endoscopy, in addition to the usual evaluation for cytomegalovirus and herpes simplex virus infections, and graft-versus-host disease. Biopsies and brushings should be obtained, and the classic ﬁndings of dichotomous hyphae with branching at angles of 45 should raise suspicion for this infection (Figure C). Because of the rare incidence of esophageal aspergillosis, no deﬁnitive guidelines for the speciﬁc treatment of esophageal aspergillosis have been published. Treatment with amphotericin B and/or voriconazole has been described in the literature, with good results. In this patient, workup for aspergillosis disseminated to other organs, which included CT, was negative, and treatment with voriconazole was initiated during his hospital stay. Antifungal therapy was continued for 2 months, at which point repeat esophagogastroduodenoscopy showed complete healing and resolution of previously seen ulcers.
References 1. 2. 3.
Eggimann P, Chevrolet JC, Starobinkski M, et al. Primary invasive aspergillosis of the digestive tract: report of two cases and review of literature. Infection 2006;34:333–338. Marr Kieren A, Carter RA, Crippa F, et al. Epidemiology and outcome of mould infections in hematopoietic stem cell transplant recipients. Clin Infect Dis 2002;34:909–917. Choi JH, Yoo JH, Chung IJ, et al. Esophageal aspergillosis after bone marrow transplant. Bone Marrow Transplant 1997; 19:293–294.
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