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A Rare Case of Syncephalastrum racemosum Invasive Pulmonary Infection after Transplantation FREE TO VIEW

Marika Gassner, DO; Hadeel Zainah, MD; Dima Arbach, MD; Hanhan Li, MD; Chad Stone, MD; Mayur Ramesh, MD; Emanuel Rivers, MD
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Henry Ford Health System, Detroit, MI

Chest. 2013;144(4_MeetingAbstracts):242A. doi:10.1378/chest.1704244
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SESSION TITLE: Infectious Disease Student/Resident Case Report Posters II

SESSION TYPE: Medical Student/Resident Case Report

PRESENTED ON: Tuesday, October 29, 2013 at 01:30 PM - 02:30 PM

INTRODUCTION: Zygomycosis is usually an opportunistic infection and often fatal in immunocompromised hosts1. Syncephalastrum species are primarily isolated in cases of onychomycosis and cutaneous infection and rarely in other locations. We describe the first case of pulmonary infection caused by Syncephalastrum racemosum .

CASE PRESENTATION: A 58-year-old female presented 59 days after living-donor liver transplantation with hyperbilirubinemia. Past history of hypertension, diabetes mellitus and liver cirrhosis secondary to hepatitis-C. She was on a standard immunosuppressive regimen and prophylaxis. Upon admission, vital signs were within normal limits. Initial laboratory studies revealed WBC 3,000 /uL, Hb 8.9 g/dL, Plt 50,000 /uL, Crea 0.95 mg/dL, ALT/AST 40/63 U/L, Bili 8.9 mg/dL and PT 18.3 sec. Physical exam showed anasarca, confusion and asterexis. On day 4, she was transferred to the ICU for acute gastrointestinal bleeding and later required mechanical ventilation and hemodialysis for hepatorenal syndrome. On day 9, she developed fevers and prolonged bacteremia with vancomycin-resistant Enterococcus faecium and Enterobacter cloacae, due to central venous line infection. Bronchoscopy revealed a necrotic lesion within the right upper bronchus. Histology of bronchial biopsy showed S. racemosum hyphae (Image 1). Bronchial lavage grew Aspergillus niger. Serum Beta-D glucan was elevated >500 pg/mL. Chest X-Ray revealed a left upper lobe cavitary lesion. CT chest confirmed a 2.5x3.3cm lesion and left lower lobe nodules (Image 2). Voriconazole and liposomal amphotericin-B (LAMB) were initiated. However on day 30 she succumbed to overwhelming sepsis and fulminant liver failure.

DISCUSSION: Syncephalastrum is a heterothallic filamentous fungus of the family Zygomycete that is found in soil and animal feces primarily in tropical and subtropical areas. S. racemosum is the only pathogenic species in this genus, often considered a contaminant, and only rarely associated with human disease. Our case represents the first reported human pulmonary infection. Histology revealed aseptate hyphae branching in wide angle characteristic of Zygomycetae. Sporangiophores terminated in swollen vesicles with radial merosporangiae filled with spores confirmed the diagnosis of S. racemosum. Aspergillus niger was likely contributing to the pulmonary disease an was responsebile for the elevated Serum Beta-D glucan . Alternative treatment to the antifungal therapy would have been surgical resection, however the grave clinical status of our patient barred this. Our patient’s poor immune status was also contributory to the lack of improvement.

CONCLUSIONS: S. racemosum is rare but potentially fatal in immunocompromised hosts, and can be identified by its staining characteristics on histology.

Reference #1: Walsh TJ, et al. "Early clinical and laboratory diagnosis of invasive pulmonary, extrapulmonary, and disseminated mucormycosis (zygomycosis). Clin Infect Dis. 2012; 54 Suppl 1:S55-60

DISCLOSURE: The following authors have nothing to disclose: Marika Gassner, Hadeel Zainah, Dima Arbach, Hanhan Li, Chad Stone, Mayur Ramesh, Emanuel Rivers

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