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PubMed1991

Deferoxamine therapy and mucormycosis in dialysis patients: report of an international registry.

Fifty-nine cases of mucormycosis in dialysis patients have been reported to the registry (25 new cases and 34 previously reported cases). The presenting forms of mucormycosis included disseminated in 44%, rhinocerebral in 31%, and other forms in 25%. The diagnosis was made during life in only 39%, while the diagnosis was discovered at autopsy in 61% of the cases. The fungus, cultured in only 36%, was always Rhizopus. The infection was fatal in 86% of cases. No known risk factors for fungal infections, eg, diabetes mellitus, liver disease, splenectomy, neutropenia, steroid therapy, or other immunosuppressive therapy, were present in 70% of patients, but 78% of patients were being treated with deferoxamine. The role played by this drug and more particularly by its iron chelate, feroxamine, in the pathogenesis of mucormycosis in these patients is underscored. Because of this risk, deferoxamine therapy in dialysis patients should be limited to severe aluminum toxicity, the deferoxamine should be given at the lowest possible dose, and dialytic methods to augment the removal of feroxamine should be studied.

Adolescent↗

PubMed1987

Intestinal mucormycosis in hemodialysis patients following deferoxamine.

Two maintenance hemodialysis patients receiving deferoxamine to chelate iron and aluminum developed intestinal mucormycosis. One patient had pulmonary mucormycosis as well. The patients lacked the usual predisposing factors to mucormycosis, ie, diabetes and acidosis, but both had liver disease. The role of siderophores such as deferoxamine in promoting certain infections is discussed with reference to this particular clinical setting.

Deferoxamine↗

PubMed1997

Pulmonary mucormycosis in diabetic renal allograft recipients.

Renal allograft recipients are prone to opportunistic infections due to their need of immunosuppression to prevent rejection. Mucormycosis is a rare opportunistic infection caused by a fungi of the order Mucorales. Risk factors predisposing to this disease include prolonged neutropenia, chelation therapy for iron or aluminum overdose, diabetes, and patients who are immunosuppressed. Life-threatening infections can occur, as this fungi has the propensity to invade blood vessel endothelium, resulting in hematologic dissemination. Early diagnosis and prompt aggressive therapy is imperative to achieve an improved outcome. We present two cases of pulmonary mucormycosis in diabetic renal allograft recipients who were treated successfully with amphotericin B and surgical resection of the lesions with preservation of their allograft function. In this era of intensified immunosuppression, we may see an increased incidence of mucormycosis in our transplant population.

Adult↗

PubMed1999

Successful eradication of mucormycosis occurring in a pulmonary allograft.

The zygomycetes are saprophytic fungi that rarely cause disease in the normal human host. In immunocompromised individuals, these organisms can cause invasive infections, collectively called mucormycosis. Mucormycosis is associated with a high mortality rate, especially in organ transplant recipients. In this report, we describe the first case of successfully treated mucormycosis involving a pulmonary allograft. Treatment consisted of surgical excision of the affected lobe and chest wall and lipid-complex amphotericin B. The lipid complex formulation permitted a prolonged course of therapy that was likely critical to eradication of the infection.

Adolescent↗

PubMed1997

Mucormycosis of the mandible after dental extractions in a patient with acute myelogenous leukemia.

Mucormycosis is a fulminant fungal infection that occurs most often in diabetic and immunocompromised patients including those with hematologic malignancies. In this case, a patient with acute myelogenous leukemia developed mucormycosis in a recent mandibular extraction site. The successful management of this patient demonstrated that early diagnosis, aggressive surgical and medical treatment and resolution of the underlying disease could improve the prognosis for survival. A case is made for the role of smoking as an initiator of mucormycosis, and treatment considerations for controlling periodontal and pulpal disease before chemotherapy are discussed.

Dental Care for Chronically Ill↗

PubMed1991

Rhinocerebral mucormycosis: use of liposomal amphotericin B.

Rhinocerebral mucormycosis is a rare but often fatal condition characterized by an aggressive necrotizing infection spreading from the nose to the paranasal sinuses, orbit and hence to the central nervous system. A case is reported in which a diabetic male with advanced mucormycosis was successfully treated by a combination of surgery, supportive therapy and liposomal amphotericin B. Liposomal delivery allows the drug to be both less toxic and more effective, and this is the first reported case of its use in rhinocerebral mucormycosis.

Amphotericin B↗

PubMed1992

Rhinocerebral mucormycosis: an unusual case presentation.

Rhinocerebral mucormycosis is a rapidly fatal fungal disease which involves the nose, paranasal sinuses, orbit and central nervous system. The fungal infection is usually secondary to immunosuppression, diabetic acidosis, or antibiotic, steroid or cytotoxic therapy. It can also occur in patients suffering from burns, malignancy and haematological disorders. Current treatment consists of correction of the underlying disorder, repeated debridement of the wound in combination with intravenous amphotericin B. This paper describes our experience with a case of rhinocerebral mucormycosis. This is an unusual case in which mucormycosis was seen in a young female where no underlying cause was found. She responded to surgical debridement in combination with intravenous amphotericin B.

Adult↗

PubMed2006

Rhinocerebral mucormycosis with orbital and intracranial extension: a case report and review of optimum management.

Rhinocerebral mucormycosis is a devastating, rapidly progressive and often fatal opportunistic fungal infection predominantly affecting individuals with underlying metabolic and/or immunological compromise. Intracranial extension of the disease has invariably been associated with mortality.We present a review of optimum management of rhinocerebral mucormycosis and a case report of sinonasal mucormycosis with intracranial and orbital extension which was treated successfully with a combination of systemic liposomal amphotericin B therapy and wide surgical debridement.

Amphotericin B↗

PubMed2000

Gastrointestinal mucormycosis complicated by arterio-enteric fistula in a patient with non-Hodgkin's lymphoma.

Gastrointestinal mucormycosis is a rare, often fatal, systemic infection found predominantly in immunocompromised patients. We report a case of gastrointestinal mucormycosis in a 53-year-old female with non-Hodgkin's lymphoma. Following her first course of chemotherapy, bowel obstruction developed as a result of mucormycosis. Despite treatment with antifungal therapy, she required a laparotomy owing to severe haemorrhage caused by mucormycosal invasion of her iliac artery. With continued antifungal treatment and further chemotherapy, she ultimately underwent reversal of her Hartmann's procedure and remains disease-free.

Amphotericin B↗

PubMed2001

Fatal rhino-orbito-cerebral mucormycosis in an apparently normal host: case report and literature review.

Fungal infections of the central nervous system (CNS) are fortunately rare but remain challenging problems occurring mostly in immunocompromised individuals, with protean manifestations, unpredictable course and unfavorable outcome in many cases despite aggressive neurosurgical intervention and recent antifungal drugs. Rhino-orbito-cerebral mucormycosis is a potentially lethal opportunistic fungal infection with rapid progression and high mortality. Its pathogenic nature becomes evident when the patient's general resistance is compromised. We present a case of an invasive rhino-orbito-cerebral mucormycosis in an apparently normal adult who initially developed mild paranasal sinusitis and later developed status epilepticus and despite an aggressive management died. Interesting clinical, neuroimaging and histological findings are described, and the possibility of fatal mucormycosis in an apparently normal host is highlighted.

Adult↗

PubMed1998

[Rhino-orbito-cerebral mucormycosis: clinical presentation].

BACKGROUND: Rhino-orbito-cerebral mucormycosis is an opportunistic rapidly progressive infection affecting almost exclusively diabetic or immunocompromised patients. CASE REPORTS: Three cases are reported. For one patient mucormycosis was the first manifestation of juvenile diabetes and the evolution was favorable. In the second case the infection affected a known diabetic patient and the clinical course was fatal. The third patient was immunocompromised, showed mild clinical features and a rapidly fatal evolution, the diagnosis being made only postmortem. CONCLUSION: These three cases illustrate the wide clinical spectrum of rhino-orbito-cerebral mucormycosis, its serious nature and difficult diagnosis.

Adolescent↗

PubMed2001

Longterm survival in acute rhinocerebral mucormycosis with giant cell arteritis and foreign body granulomas.

A case of rhinocerebral mucormycosis occurring in a 41-year-old man with insulin-treated diabetes mellitus is reported. Microscopically, biopsy samples obtained from the left ethmoid and middle turbinate sinuses contained fungi that formed mycotic granulomas associated with multinucleate giant cell arteritis. The multinucleate giant cells contained broad, infrequently septate hyphase consistent with mucormycosis. The patient received surgical debridement with extenteration of the left orbit, and intravenous liposome-encapsulated amphotericin B. After 12 months, examination of the patient revealed complete healing. Multinucleate giant cell granulomas and arteritis are only exceptionally associated with rhinocerebral mucormycosis, but these histologic findings may be correlated with a progressive disease with better prognosis.

Acute Disease↗

PubMed2004

Fatal strokes in patients with rhino-orbito-cerebral mucormycosis and associated vasculopathy.

Within a 10-y period, fatal strokes occurred during parenteral administration of amphotericin B and surgical debridement of paranasal sinuses in 6 pathologically verified cases of rhino-orbito-cerebral mucormycosis (ROCM). All patients had unnoticed type-2 diabetes mellitus without ketoacidosis. They presented with unilateral orbital cellulitis and cavernous sinus syndrome. Fatal malignant cerebral infarctions occurred in the carotid system in 5 patients, and in the basilar artery or its major branches in 2 patients. Accelerated thrombotic occlusion of the cavernous portion of the carotid artery or the basilar artery was likely to be due to mucormycosis associated-vasculopathy and diabetic vasculopathy. One patient died of massive subarachnoid hemorrhage following rupture of the mycotic aneurysm. Despite parenteral administration of amphotericin B, fatal outcome of ROCM in patients with unnoticed diabetes mellitus occurs due to mucormycosis-associated malignant strokes. To improve outcome, a combination of early radical debridement, ocular exenteration, parenteral and local administration of amphotericin B, and decompression craniotomy should be considered.

Aged↗

PubMed2003

Eradication of invasive mucormycosis--effectiveness of the Echinocandin FK463.

BACKGROUND: Invasive rhinocerebral mucormycosis is a rare and often fatal opportunistic fungal infection. It is encountered in immunocompromised hosts exemplified by those with diabetes, human immunodeficiency viruses and particularly haematologic malignancies typically after high-dose chemotherapy and stem cell transplantation. In contrast to the more usual outcome with rapid progression and death. We now describe a successful eradication attributable to the use of a newly available antifungal agent. SETTING: Haematology department and bone marrow transplantation unit. MATERIAL AND METHOD: Two patients are contrasted. The first with acute leukaemia developed rapidly progressive facial swelling with mucormycosis proven on biopsy. Treatment over 2 months with maximally tolerated doses of amphotericin failed to halt intracranial extension and death resulted. The second, presented with acute lymphoblastic leukaemia in August 1997, underwent successful autologous bone marrow transplantation in February 1998. Relapse followed in March 1999 and after reinduction and consolidation receive a matched unrelated volunteer allograft in September 1999. A second recurrence was documented in April 2000 and in spite of achieving remission he developed a fever that was managed empirically with intravenous amphotericin and, on discharge, oral itraconazole. Left-sided facial swelling expanded rapidly and biopsy showed extensive invasion of the maxillary sinus with mucormycosis. FK463 was added on 5 June 2000 with gradual reduction in facial pain and within 1 month all clinical signs and resolved. Serial biopsies that included histopathologic investigation and microbiologic cultures confirmed eradication of the invasive mucor. In view of the potential danger of recrudescence this treatment regimen was continued through further chemotherapy and, once again disease-free, a second matched unrelated volunteer allograft took place in August 2000. Full reassessment at the time failed to demonstration any residual fungus. Engraftment was confirmed but neutropenic sepsis resulted in severe inflammatory response syndrome with progression to multiple organ dysfunction to which he succumbed without any evidence of leukaemic or systemic mycosis. CONCLUSION: Echinocandin FK463 is of documented value in managing invasive candidiasis and aspergillosis. This is believed to be the first case of successful outcome with one of the angiotrophic zygomycetes.

Acute Disease↗

PubMed1998

Rhinocerebral mucormycosis treated with amphotericin B colloidal dispersion in three patients.

Rhinocerebral mucormycosis (zygomycosis) primarily affects diabetic or immunosuppressed patients and typically progresses rapidly, necessitating surgical excision and antifungal therapy with amphotericin B. Large doses of amphotericin B are required for cure, causing significant renal toxicity. Amphotericin B colloidal dispersion (ABCD; Amphocil, Sequus Pharmaceuticals, Menlo Park, CA) is a 1:1 complex of cholesteryl sulfate and amphotericin B, which results in significant reduction of toxicity, especially nephrotoxicity. We describe three patients with life-threatening rhinocerebral mucormycosis treated with ABCD. All patients had high serum creatinine levels due to prior treatment with amphotericin B; these levels reverted to normal during treatment with ABCD. Two patients with diabetes mellitus were cured after receiving a combination of surgery and ABCD therapy. The third patient, who had myelodysplastic syndrome, had an initial good response, with cure of the fungal infection; however, he eventually died of his primary illness. To the best of our knowledge, this is the first detailed clinical description of the treatment of mucormycosis with ABCD.

Adult↗

PubMed1975

Mucormycosis in a transplant recipient.

Mucormycosis classically occurs in patients who have uncontrolled diabetes who develop rhinocerebral disease. A fatal case of rhinocerebral infection caused by Rhizopus arrhizus in a 53-year-old man who had received a renal homograft three years previously is reported. Only five similar cases have been documented, all since 1970. Although direct smears of the purulent nasal exudate revealed the presence of numerous Gram-negative bacilli, later identified as Haemophilus influenzae, the diagnosis of mucormycosis was made by demonstrating the typical broad, nonseptate branched hyphae in the necrotic tissue obtained by surgical debridement of the paranasal sinuses. Culture of this material revealed growth of mold-like fungus which, upon direct microscopic examination, showed sporangiophores bearing spherical sporangia arising directly from a cluster of root-like structures of rhizoids. Despite the immediate institution of therapy with amphotericin B postoperatively, the patient died 48 hours later. Subsequently, the Rhizopus isolated was shown to be resistant to both amphotericin B and 5-fluorocytosine. The present case and two others stress the importance of an aggressive diagnostic approach to patients suspected of having mucormycosis, because the usual microbiologic technics are frequently, inexplicably, unsuccessful, and possibly even misleading in this disease.

Adult↗

PubMed1987

Unusual histopathology of mucormycosis in acute myelogenous leukemia.

Mucormycosis is a mycotic disease caused by certain genera of the class Zygomycetes (order Mucorales). It is usually a rapidly progressive opportunistic infection characterized by vascular invasion, neutrophilic response, and intravascular thromboses. Entomophthoromycosis describes another disease caused by Zycomycetes, but of the order Entophthorales. In contrast to mucormycosis, entomophthoromycosis is not associated with underlying disease. Vascular invasion by the hyphae does not occur, and the hyphae are surrounded by eosinophilic sheathes. These sheathes are regarded as one form of the Splendore-Hoeppli phenomenon. The authors report an unusual case of mucormycosis in a severely pancytopenic patient dying of acute myelogenous leukemia. At autopsy, eosinophilic sheathes with radiating fibrils were seen about intravascular hyphae, simulating entomophthoromycosis. This pseudo-Splendore-Hoeppli phenomenon appears to be caused by adsorption of plasma and fibrin to the hyphal walls in the absence of both neutrophilic response and extensive fibrin clotting.

Aged↗

PubMed1994

Mucormycosis: emerging prominence of cutaneous infections.

Twenty-five patients with mucormycosis were seen at two university-affiliated hospitals from 1979 to 1993. These cases included 10 cutaneous, 9 rhinocerebral, and 3 disseminated infections, as well as one case each of pulmonary, renal, and peritoneal dialysis catheter-related infection. Eleven of the patients were diabetic and seven had ketoacidosis, including four who became acidotic after admission to the hospital. The mortality rates associated with rhinocerebral, disseminated, and pulmonary infections were 78%-100%, while those associated with cutaneous and miscellaneous forms were zero. In view of the prominence of cutaneous infections, the 10 cases of cutaneous mucormycosis (in addition to a case from a community hospital) are reported in detail. Systemic diseases were present in four of the 11 patients. Local factors leading to infection were identified in nine of the cases and included motor vehicle accident-related and other trauma, surgery, a spider bite, and an intravenous infusion catheter. The cases of cutaneous mucormycosis reported in the literature have been analyzed for identification of predisposing factors, treatment, and outcome. Aggressive surgical debridement is the most important component of therapy, and administration of amphotericin B is a useful adjunct. Skin grafting is useful as a method of repairing defects left by extensive debridement.

Adolescent↗