Fulminant zygomycosis of graft liver following liver transplantation

Abstract

A 44-year-old man with hepatitis B virus (HBV)-related cirrhosis underwent living donor liver transplantation at our institute. Induction of immunosuppression was achieved with basiliximab, due to deranged renal function, and maintained with prednisolone, tacrolimus and mycophenolate mofetil. The intraoperative and immediate postoperative periods were fairly uneventful. A duplex scan, taken during the third week post-transplantation due to sudden rise in liver enzymes, revealed multifocal hypoechoic lesions in the graft liver with normal Doppler parameters. Multidetecor computed tomography (MDCT) showed multiple hypodense vessel-sparing lesions in the graft liver. Cultures from the aspirate grew filamentous fungi identified as Basidiobolus ranarum species. Despite multiple broad spectrum antifungal infusions including liposomal amphotericin, itraconazole, caspofungin and posaconazole, serial sonography showed the hepatic lesions increasing in size, and involving segments V, VI and VII. The patient developed severe liver dysfunction ultimately progressing to sepsis, multiorgan dysfunction and death.

Gastrointestinal basidiobolomycosis: An emerging mycosis difficult to diagnose but curable. Case report and review of the literature

“…Although a definite diagnosis can be obtained with culture [14,15,18,19,[22][23][24]30,33,35,41,45,47,[49][50][51]54,59,62], it was frequently missed in previous reports because of the lack of suspicion that made tissue specimens unavailable for culture purpose. Therefore, the diagnosis of gastrointestinal infection by B. ranarum was mainly obtained on histologic examination, which typical morphologic features include granulomatous inflammation and a diffuse eosinophilic infiltrate with thin walled branched hyphae surrounded by eosinophilic material (Splendore-Höeppli phenomenon) and sometimes zygospores (spherical bodies with foamy cytoplasm) [4,6,12,16,17,20,24,[26][27][28][29][31][32][33][35][36][37][38][39][42][43][44]46,49,52,53,…”

“…However, as suggested by Bering et al, the absence of demonstrable in vitro activity of KI against B. ranarum coupled with several possible limitations (toxicity associated with high doses, lack of any standard prescription recommendation, available new azoles) do not recommend its use in GIB [68]. More recently successful treatments with voriconazole [42,49,50,[53][54][55]58,59] and posaconazole [40,48,50] have been reported. On the basis of the experience reported in the literature azoles should be considered the drugs of choice for GIB.…”

“…A male prevalence was observed (76/102, 74.5%) with a 3:1 ratio. All but one patient were immunocompetent [59]. The median age was 19 years (range 1-81 years) and 42 patients (41.2%) were children (age 1-13 years).…”

“…decades a 15-fold increase in cases of gastrointestinal infection by B. ranarum has been reported worldwide. Although most of the cases have been described in the Middle East (Saudi Arabia, Iran, Iraq, Kuwait, Oman, Qatar) [23-26,29,31-39,41-46,48-50,52-55,57,60,62] and the southwestern of United States (Arizona and Utah)[15,[17][18][19][20]40,47,56], sporadic reports came from South America, Africa, Europe and the Indian Subcontinent[4,12,14,16,27,28,30,51,59,PR] and no clear environmental risk factors have been identified. In the case-control study performed by Lyon et al in Arizona, ranitidine use (OR 6.0) and a longer period of smoking (OR 2.1/additional 20 years of smoking) were both associated with the development of the disease…”

Liver and Bile Duct Infections

“…368 The diagnosis is often not made B A allografts leading to retransplantation has been described. 387,388 Less commonly, it may affect immunocompetent patients. 389 Hepatic infection in the absence of pulmonary disease may be caused by spread from a gastrointestinal source, and thus many patients have concurrent involvement of a site in the tubular gastrointestinal tract.…”