Case study Candida albicansassociated necrotizing vasculitis producing life-threatening gastrointestinal hemorrhage Jeremy Sargent MD a , Aengus O'Marcaigh MD a , Owen Smith MD a , Karina Butler MD b , Patrick Gavin MD b , Maureen OSullivan MD, PhD c, a Department of Hematology, Our Lady's Children's Hospital, Crumlin, Dublin 12, Ireland b Department of Infectious Disease, Our Lady's Children's Hospital, Crumlin, Dublin 12, Ireland c Department of Pathology, Our Lady's Children's Hospital, Crumlin, Dublin 12, Ireland Received 21 July 2009; revised 1 September 2009; accepted 15 September 2009 Keywords: Candida; Vasculitis; Fibrinoid necrosis Summary Patients undergoing treatment of acute lymphoblastic leukemia are at risk for fungal infections including disseminated candidiasis. We describe a case of systemic Candida albicans infection associated with life-threatening gastrointestinal hemorrhage due to unusual necrotizing vasculitis involving the gastrointestinal tract. We explore the association between Candida and such vasculopathy. © 2010 Elsevier Inc. All rights reserved. 1. Case report A 3-year-old girl presented with spontaneous bruising and lethargy and was diagnosed with precursor B lymphoblastic leukemia and commenced on induction chemotherapy comprising dexamethasone, vincristine, polyethylene glycol (PEG)-asparaginase, and intrathecal methotrexate. She was admitted on day 20 with fever and neutropenia, initially treated with piptazobactam, gentamicin, and oral nystatin with subsequent addition of teicoplanin. After 1 week of unresolved pyrexia, an ultrasound of abdomen showed hepatomegaly with no focal lesions and a splenic lesion (4 × 4 mm). Candida albicans was isolated on culture of urine. Liposomal amphotericin (5 mg/kg per day) was initiated. For the next 4 days, she remained febrile and developed tender abdominal distension with progressive hepatosplenomegaly and new-onset melena with a fall in hemoglobin level from 86 g/L to 53 g/L. A repeat ultrasound of abdomen revealed a single hepatic lesion and 3 splenic lesions. Computed tomogram of the abdomen 10 days later showed multiple hepatic, splenic, bilateral renal, and cerebral lesions. Over the ensuing 3 weeks, she continued to have significant melena and fresh bleeding per rectum. This was managed medically in the intensive care unit. Endoscopy of the upper gastrointestinal tract showed gastric antral ulcers and a large duodenal ulcer, which was injected with adrenaline. Patchy erosions were identified from the duodenum through jejunum. Because of uncontrollable ongoing hemorrhage, a laparotomy was performed. At this stage, she had had 21 days of systemic antifungal therapy including liposomal amphotericin and caspofungin. Hepato- megaly with liver abscess formation was noted at surgery, and the abscess was sampled and a wedge biopsy of liver taken also. Candida albicans only was cultured from the abscess contents. Examination of the small intestine showed multifocal penetrating ulcers, and a 10-cm section of jejunum was resected. The histologic findings (discussed in detail below) were of a necrotizing vasculitis. Serum antineutrophil cytoplasmic antibodies were not detected. High-dose methylprednisolone (initially 30 mg/kg daily for 3 days then 5 mg/kg daily with gradual tapering) was commenced. Corresponding author. www.elsevier.com/locate/humpath 0046-8177/$ see front matter © 2010 Elsevier Inc. All rights reserved. doi:10.1016/j.humpath.2009.09.015 Human Pathology (2010) 41, 602604