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Rhino-orbital and rhino-orbito-cerebral mucormycosis.

BACKGROUND: Rhino-orbito-cerebral mucormycosis (ROCM) is a devastating infection of immunocompromised hosts. We present our experience with 19 ROCM cases and attempt to define preferred diagnostic and treatment protocols. METHODS: All had tissue biopsies obtained studied by direct smear, histologic studies, and cultures. Imaging was obtained in 14 cases. RESULTS: Sixteen patients presented between August and November. Six had mixed fungal infections. Seven patients had end-stage underlying disease or infection and did not undergo surgery and 4 had an indolent form of disease. Patients were treated by surgery and by amphotericin B. The overall survival was 47%. CONCLUSIONS: ROCM may have seasonal incidence peaking in the fall and early winter. The therapeutic approach should be unchanged in cases of mixed fungal infections. Amphotericin B with aggressive debridement remains the mainstay of treatment. Early recognition and treatment are essential. A presentation and survival-dependent classification of ROCM are offered.

Adult↗

Atypical cause of forearm skin ulceration in a leukaemic child: mucormycosis. A case report.

Primary cutaneous mucormycosis is an uncommon, deep and aggressive fungal infection occurring mainly in immunosuppressed or diabetic patients. Rapid diagnosis and therapy are necessary to prevent a fatal outcome. An eight-year-old leukaemic child presented with a dark necrotic ulcer on the volar-ulnar aspect of the left forehand. The lesion had developed over seven days, beginning as a vasculo-haemorrhagic erythematous plaque. There was no known history of trauma to the area, but skin necrosis from external compression caused by an intravenous line could not be completely ruled out. The lesion rapidly progressed to a 5 x 8 cm painful necrotic ulcer with an erythematous border. Treatment with ichthyol dressing and intra-venous antibiotherapy failed to improve the condition. Wide debridement was performed and specimens were sent for microbiology and pathology examinations. Microscopic examination demonstrated broad, irregularly walled, non-septate fungal hyphae that were consistent with Mucor. Amphotericin B was administrated intravenously (1 mg/kg/day) and hydrogel and hydrophile adhesive polyurethane foam dressings were applied. After 12 days, the soft tissue defect was covered with a split-thickness skin graft, harvested from the lateral aspect of the thigh. Diagnosis of this infection is based on complete histopathological and microbiological studies. Awareness, and a high index of suspicion are required because of the potential fulminant and fatal course.

Amphotericin B↗

Usefulness of frozen section in rhinocerebral mucormycosis diagnosis and management.

AIMS: Rhinocerebral mucormycosis (RCM) is a well-described fulminant fungal infection that presents acutely in patients with ketoacidosis and immunosuppression. Very early diagnosis, established with the demonstration of characterised hyphae in tissues, greatly improves the prognosis of RCM. In this regard, the specificity and the sensitivity of frozen section for the diagnosis and the surgical debridement of RCM were evaluated in this study. METHODS AND RESULTS: Frozen section was performed for the diagnosis (six of seven cases) and surgical treatment (three of seven cases) of RCM. In all cases, diagnosis was made by frozen section and confirmed by histological examination. Frozen section allowed radical surgical excision of infected tissue. In all cases, invasive, broad-based non-septated hyphae with branching at right angles were well demonstrated on toluidine blue staining. Cultures were positive for Rhizopus oryzae in three of seven cases. Outcome was favourable for five of seven patients and two patients died after the diagnosis. CONCLUSIONS: Frozen section is a specific and sensitive method to make both a quick initial diagnosis of RCM and to successfully eradicate the tissue infected by organisms belonging to the order Mucorales.

Adult↗

Murine model of pulmonary mucormycosis in cortisone-treated mice.

Intranasal inoculation of Rhizomucor pusillus sporangiospores into cortisone-treated mice produced pulmonary and disseminated mucormycosis (phycomycosis). Evidence for infection in cortisone treated mice was obtained by recovery of Rh. pusillus from homogenates of tissue. Confirmation of infection was shown histologically. The 50% infectious dose was 2.4 x 10(2) colony forming units for lung infections and 2.7 x 10(5) colony forming units for brain infections. No evidence of sporangiospore germination was found in tissues of non-cortisone-treated mice although sporangiospores were found throughout pulmonary tissues. In infected tissues of cortisone-treated mice, hyphae were covered with leukocytes and tissue necrosis was extensive.

Animals↗

Liposomal amphotericin B and surgery in the successful treatment of invasive pulmonary mucormycosis in a patient with acute T-lymphoblastic leukemia.

Pulmonary mucormycosis is a usually fatal opportunistic infection in immunocompromised patients. We describe the first case of an adult patient with hematological malignancy and profound neutropenia to survive a disseminated pulmonary Rhizomucor pusillus infection. Early diagnostic procedures combined with high doses of liposomal amphotericin B and surgical resection may have contributed to the successful outcome.

Amphotericin B↗

Subacute pulmonary mucormycosis.

This report describes two patients with subacute pulmonary mucormycosis who died of pulmonary hemorrhage. Both patients had diabetes mellitus, but neither patient had the typical fulminant illness associated with this infection. The subacute progressive illness seen in these patients is unusual and suggests the need for aggressive diagnostic and therapeutic measures in patients who are at risk for this infection.

Diabetes Complications↗

Cutaneous mucormycosis in a young, immunocompetent girl.

We report a case of cutaneous mucormycosis in a healthy, immunocompetent young girl (age 14 years). The patient had a 5-year history of a slowly enlarging, erythematous plaque with slight elevated, scaling, circinate borders on the right thigh. Histopathology showed a granulomatous infiltrate with broad, pale, non-septate hyphae. Mycological study identified Mucor hiemalis (Wehmer).

Adolescent↗

A Rhizopus oryzae strain isolated from resected bone and soft tissue specimens from a sinonasal and palatal mucormycosis case. Report of a case and in vitro experiments of yeastlike cell development.

We report a histologically and mycologically proven sinonasal mucormycosis case causing palatal necrosis in a nondiabetic patient with renal failure. Mycological examination of Giemsa stained imprinted tissue preparations revealed abundant yeast-like cells besides the typical mucoraceous hyphae. The fungus was isolated from surgical specimens and identified as Rhizopus oryzae by phenotypic and genotypic tests. Laboratory studies were performed to investigate the association of the yeast-like cells observed in tissue specimens and the fungus recovered in culture. In vitro induced yeast-like cell development of the case isolate was found under certain growth conditions and documented by photomicrographs.

Bone and Bones↗

The development of the acute inflammatory response to experimental cutaneous mucormycosis in normal and diabetic rabbits.

The histologic changes associated with the development of the acute inflammatory response to experimental cutaneous mucormycosis were studied at various times from 5 minutes to 24 hours after inoculation into normal rabbits and in rabbits with acute alloxan diabetes and acidosis. In normal animals the response by polymorphonuclear leukocytes began within a few minutes after inoculation, increased rapidly in extent and intensity and reached its peak within 6 to 12 hours. By that time there was also early proliferation of fibroblasts and of large mononuclear cells and these cellular reactions, together with the accumulated granulocytes, began to produce a demarcation of the lesions. Beginning 4 to 6 hours after inoculation the fungus showed some growth but this remained confined to the necrotic center of the lesions. In the diabetic rabbits the onset of the response by polymorphonuclear leukocytes was delayed by several hours, reduced in intensity and was apparently less effective. There was no proliferation of fibroblasts and the lesions were spreading rather than circumscribed. Fungus growth in the tissues began shortly after inoculation, was marked, progressed rapidly, and soon extended beyond the site of inoculation. The large mononuclear cells, however, appeared at about the same time and in equal number in the lesions of both diabetic and non-diabetic animals and showed no morphologic changes. It is concluded that a significant delay and impaired effectiveness of the response by polymorphonuclear leukocytes, a lack of fibroblastic proliferation and an enhanced growth of the fungus lower host resistance to infection with it and are directly consequent on severe alterations in host metabolism.

Animals↗

Complete video-assisted resection of trilobar mucormycosis.

A case of trilobar pulmonary mucormycosis in a diabetic patient with severe obstructive pulmonary disease, successfully treated with systemic antifungal therapy and complete video-assisted thoracic surgery (VATS) resection, is presented. The VATS approach permitted accurate diagnosis and definitive therapy using lung-sparing techniques in a minimally invasive manner.

Humans↗

Disseminated mucormycosis in a case of metastatic carcinoma.

A 50-year-old white woman who underwent bilateral mastectomy for fibrocystic disease of the breast in 1974 and laminectomy for lumbar disc fusion in 1979, was diagnosed early in 1980 at Roswell park Memorial Institute as a case of metastatic anaplastic carcinoma. She was started on platinum combination therapy, but in the course of her treatment she became increasingly jaundiced, then semi-comatose and died suddenly two months after the diagnosis of carcinoma. Histopathologic examinations revealed undifferentiated small-cell carcinoma in the lung, liver, lymph nodes, and bone. Mucormycosis, characterized by the presence of broad coenocytic hyphae associated with thrombosis, infarction, and necrosis, was unexpectedly discovered at postmortem examination in the brain, lung, and heart. The etiologic agent, Absidia corymbifera, was isolated from a lung specimen and was identified by its morphologic characteristics and by the formation of zygospores in crosses with a compatible tester strain of the mating type (-).

Carcinoma, Small Cell↗

Cunninghamella: a newly recognized cause of rhinocerebral mucormycosis.

Cunninghamella, a zygomycete in the order Mucorales, is an extremely rare cause of human infection. Of the five reported cases of human disease caused by this fungus, none involved rhinocerebral infection. Here, the authors document what appears to be the first case of rhinocerebral mucormycosis caused by Cunninghamella bertholletiae in an elderly man who had diabetes with sideroblastic anemia and hemochromatosis. The disease was rapidly fatal. The mycology and classification of this organism are presented, and the previous case reports in the literature are reviewed.

Aged↗

Widely disseminated Cunninghamella mucormycosis in an adult renal transplant patient: case report and review of the literature.

A case of widely disseminated mucormycosis involving Cunninghamella bertholletiae is reported. This represents the first report of Cunninghamella infection in a transplant patient and, to our knowledge, only the eighth reported case of human infection by this saprophytic fungus. The clinical course was similar to most of the previously reported cases in that the patient was immunocompromised prior to the infection and the diagnosis was made too late for appropriate therapeutic intervention. The current case is unique in that it involved a renal allograft recipient; that the major clinical signs and symptoms mimicked myocardial infarction; and that there was more widespread dissemination than in earlier cases. The histologic and laboratory identification of the fungus are presented and correlated with the clinical parameters (signs, symptoms, and therapy) of this case as well as the cases previously reported in the literature.

Adult↗

Hospital-acquired wound mucormycosis.

Cutaneous infections due to fungi of the order Mucorales are uncommon and usually present as a fulminant necrotizing cellulitis. We describe a case of a progressive wound infection at a surgical drain site caused by Rhizopus rhizopodoformis. The indolent nature of the infection and lack of systemic toxicity were atypical features. Mucormycosis should be suspected in cases of slowly progressive cellulitis in the appropriate clinical setting.

Cross Infection↗

Hepatic mucormycosis in a bone marrow transplant recipient who ingested naturopathic medicine.

This case report describes a bone marrow transplant recipient in whom hepatic zygomycosis developed after ingestion of multiple naturopathic medicines. Mucor was isolated from the patient's liver aspirate and from one of the naturopathic medicines. Arbitrary-primed polymerase chain reaction (PCR) analyses were performed on the Mucor isolates from the patient's liver aspirate and from his naturopathic medicine to see if they were genotypically related. Mucor indicus was the species identified in both the patient's liver aspirate and the naturopathic medicine. Arbitrary-primed PCR analysis revealed that these isolates were genotypically identical. We conclude that this bone marrow transplant recipient acquired hepatic mucormycosis from ingestion of a naturopathic medicine containing Mucor.

Adult↗

Neonatal colonic mucormycosis--a tropical perspective.

Neonatal gastrointestinal (GI) mucormycosis is a rare, usually fatal, opportunistic fungal infection, which is difficult to diagnose early or preoperatively. We report three babies, only one of whom survived, with a review of the literature. All three had similar findings of necrosis of colon with multiple perforations. While the first baby was diagnosed as long segment Hirschsprung's, the second was treated as small left colon but went on to show signs of peritonitis. The third presented with pneumonia, which progressed to sepsis and peritonitis. All three were diagnosed by histology postoperatively and two of them succumbed, one in spite of amphotericin and the other as he was too sick to start antifungals and had a rapid downhill course. The one who survived did so even though she did not receive amphotericin, but had clear margins of resection. The only chance of survival in this fatal disease is early diagnosis and rapid institution of aggressive therapy inclusive of adequate surgical debridement and appropriate antifungal medications.

Biopsy, Needle↗

Nosocomial pulmonary mucormycosis with fatal massive hemoptysis.

We postulate that the previously healthy woman reported here developed abnormal host defense mechanisms because of acute renal failure, metabolic acidosis, hyperglycemia, and glucocorticosteroid administration. Pneumonia unresponsive to antibiotics terminated in massive fatal hemoptysis that was due to mucormycosis with rupture of the pulmonary artery into the tracheobronchial tree.

Acute Kidney Injury↗