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PubMed2005

[Imaging of rhinocerebral mucormycosis].

PURPOSE: Rhinocerebal mucormycosis is a rare life threatening fungal infection observed in immunocompromised patients. We report six cases of patients with rhinocerebral mucormycosis confirmed histologically. Our study confirms the necessity of early diagnosis when clinical and CT findings are suggestive. MATERIALS AND METHODS: This is a retrospective study including 6 diabetic patients (3 women and 3 men) aged from 28 and 63 years. Five patients had ethmoiditis evolving for a few days (3 to 5 days), and one patient was in an ketoacidotic coma and had a severe infectious syndrome with purulent rhinorrhea evolving for 4 days. All of our patients underwent computed tomography (CT) scan of the paranasal sinuses. MRI was performed in two patients with neurological findings. RESULTS: Unilateral ethmoido-maxillary sinusitis was noted in 5 cases. Only one case of pansinusitis was found. All patients presented orbital involvement. Cerebral involvement was noted in 4 cases (cerebral venous thrombosis: 2 cases; abscess: 2 cases; cerebral ischemia: 2 cases). The diagnosis of mucormycosis was based on endonasal biopsy. When available, MRI allowed a more precise evaluation of the orbital and cerebral extension. CONCLUSION: Mucormycosis is an opportunist mycosis due to mucoralis fungus. It is very invasive with a highly aggressive potential in diabetic or immunocompromised patients. Imaging study particularly CT scan, plays an important role in diagnosis especially to evaluate cerebral extension.

Adult↗

PubMed2006

Mucormycosis in the Eastern Mediterranean: a seasonal disease.

Mucormycosis is a fairly rare fungal infection caused by ubiquitous fungi of the order Mucorales and primarily affects immunocompromised hosts. A series of 16 cases of invasive mucormycosis admitted to three referral centres in Beirut, Lebanon between 1981 and 1999 is described. It includes 12 patients with rhinocerebral, three with cutaneous, and one with pulmonary infection. Onset of symptoms occurred in the summer and autumn in 15 out of 16 patients, showing a statistically significant seasonal variation (P=0.007) A recent report of 19 patients from Tel Aviv describes a strikingly similar seasonal pattern. Studies on atmospheric concentration of Mucorales spores in the Eastern Mediterranean are lacking. Weather pattern analysis in Beirut revealed clustering of onset of invasive mucormycosis at the end of a dry, warm period, which begins around May and ends in October. Mucormycosis incidence appears to be seasonal in the Eastern Mediterranean.

Adolescent↗

PubMed1999

Mucormycosis in allogeneic bone marrow transplant recipients: report of five cases and review of the role of iron overload in the pathogenesis.

In a 10-year consecutive series of 263 allogeneic bone marrow transplant recipients, we identified five cases (1.9%) of invasive mucormycosis. Only one infection occurred within the first 100 days after transplantation, while the remainder complicated the late post-transplant course (median day of diagnosis: 343). Sites of infection were considered 'non-classical' and included pulmonary, cutaneous and gastric involvement. No case of fungal dissemination was observed. Mucormycosis was the primary cause of death in three of the five patients. Corticosteroid-treated graft-versus-host disease, either acute or chronic, or severe neutropenia were present in all cases. However, compared with a matched control population, the most striking finding was the demonstration of severe iron overload in each of the mucormycosis patients. The mean level of serum ferritin, transferrin saturation and number of transfused units of red cells (2029 microg/l, 92% and 52 units, respectively) in the study group is significantly higher compared with the control group (P < 0.05). The difference with other risk groups for mucormycosis, including deferoxamine-treated dialysis patients and acidotic diabetics, was analyzed in view of the possible pathogenic role of iron. Although these infections are often fatal, limited disease may have a better prognosis if diagnosed early and treated aggressively.

Adult↗

PubMed2002

Primary cutaneous mucormycosis in infants and neonates: case report and review of the literature.

A case of angioinvasive cutaneous mucormycosis in a premature infant, eventually requiring extracorporeal membrane oxygenation therapy, is described. The fungal infection began at the site of a brachial artery catheter that had been covered with an adhesive dressing in the left antecubital fossa. The infection progressed rapidly over a 5-day period, and a left arm amputation was required. Fungal hyphae were present at the margins of resection. The patient eventually had disseminated mucormycosis and died. A second case of cutaneous mucormycosis in another premature infant also is presented. This infant had the infection at an intravenous catheter site. Rapid initiation of surgical debridement of the wound and amphotericin B therapy resulted in patient survival. Eighteen reported cases of cutaneous mucormycosis in neonates were found and are reviewed. Prematurity, low birth weight, broad-spectrum antibiotics, corticosteroid therapy, and local trauma to the skin site were common risk factors. Only 7 of the 18 patients survived. Therapy consisted of local debridement and intravenous amphotericin B. High index of suspicion, early diagnosis, and rapid institution of therapy can improve survival rate. The key to prevention appears to be appropriate skin care.

Catheterization, Peripheral↗

PubMed2000

[Computed tomographic appearance of pulmonary mucormycosis].

AIM: Analysis of the morphological characteristics of pulmonary mucormycosis using computed tomography (CT). MATERIAL AND METHODS: Prospective analysis of CT studies in 9 patients out of 19 patients with proven pulmonary mucormycosis. RESULTS: Pulmonary mucormycosis was most frequently found in the upper lobe bilaterally (51% of all nodules). In 22% of the manifestations, the morphological criterion of a "bird's nest" could be verified in CT, in 37% central necrotic areas were detected. In 37% an open bronchus was diagnosed, in 6% we observed bronchiectases. 43% of all nodules presented with a halo sign. In three patients we found pleural effusions, enlarged mediastinal lymph nodes were diagnosed in one patient. CONCLUSION: Computed tomography allows an improved diagnosis and therapeutic follow-up in patients suffering from pulmonary mucormycosis.

Adult↗

PubMed2006

Localized bilateral paranasal mucormycosis: a case in an immunocompetent patient.

Mucormycosis of the nasal cavity and paranasal sinuses is an uncommon opportunistic fungal infection, which often has an aggressive, life-threatening course. Patients who have this condition are generally diabetic or immunosuppressed. However, mucormycosis can also occur in immunocompetent individuals. The most effective treatment consists of reversal of the source of immunocompromise, immediate surgical debridement and administration of systemic amphotericin B. No consensus has been reached regarding the appropriate surgical treatment or the total dose of amphotericin B. We present the case of a patient suffering from localized bilateral paranasal mucormycosis who was treated by means of endoscopic sinus surgery and administration of systemic amphotericin B. We suggest that endoscopic sinus surgery is the choice of treatment for localized paranasal mucormycosis in an immunocompetent patient, and that the total dose of amphotericin B can be determined by the extent of disease and the postoperative endoscopic findings.

Aged↗

PubMedDate not supplied

Cardiac mucormycosis in a child with severe aplastic anemia: a case report.

Mucormycosis is an uncommon, severe, life-threatening fungal infection in the immunocompromised host. Mucormycosis with aplastic anemia is seen rarely. Only a few cases of cardiac mucormycosis with aplastic anemia have been reported in the literature. The authors present a case with severe aplastic anemia that did not respond to classic and immunosuppressive treatment for disease and developing invasive cardiac mucormycosis despite empiric treatment for febrile neutropenia.

Adolescent↗

PubMed2004

Disseminated mucormycosis associated with invasive pulmonary aspergillosis in a patient treated for post-transplant high-grade non-Hodgkin's lymphoma.

The incidence of mucormycosis, defined as systemic infection caused by fungi of the class Phycomycetes has been increasing over the past 2 decades, especially in profoundly immunocompromised hosts. We report a new case in a patient presenting with post-transplant high-grade non-Hodgkin's lymphoma who received a prolonged treatment with voriconazole and caspofungin for an invasive pulmonary aspergillosis. Definite diagnosis of mucormycosis was made by liver biopsy of nodules mimicking progressive lymphoma. The patient died 1 week after the diagnosis of mucormycosis despite the administration of liposomal amphotericin B. The role of voriconazole and caspofungin in the emergence of mucormycosis is discussed.

Amphotericin B↗

PubMed1982

Cardiac mucormycosis. A report of five patients and review of 14 previously reported cases.

Five patients with mucormycosis of the heart are described. Two had leukemia,, one had multiple myeloma, one sideroblastic anemia, and one had been wounded when a body trap exploded. None had diabetes. In addition to heart involvement, each had mucormycosis of the lungs. Kidney was infected in two patients and gastrointestinal tract, spleen, liver, adrenal, brain, and skin were each involved in one patient. Three patients had cardiac symptoms. The findings in ten previously reported patients with de novo cardiac mucormycosis and in four patients with mucormycosis occurring in association with cardiovascular surgery are summarized.

Adult↗

PubMedDate not supplied

Rhinocerebral mucormycosis in renal transplant recipients: report of three cases and review of the literature.

Mucormycosis is an opportunistic infection caused by fungi of the order Mucorales. The commonest clinical form is rhinocerebral mucormycosis, which has been described as characteristically complicating diabetes mellitus and leukemia. Three patients with rhinocerebral mucormycosis complicating renal transplantation are described, and 11 additional cases recorded in the English-language medical literature are reviewed. The mean age of the 14 patients was 36 years, and the ratio of males to females was 1.8:1. Diabetes mellitus was present in only five patients, and polycystic kidney was the most common underlying renal disease. Most kidney grafts were obtained from cadavers. Eight patients had evidence of graft rejection, and the majority had been receiving corticosteroids and azathioprine. The initial manifestations of infections became evident two days to four years after transplantation (median, two months). Facial swelling, tissue necrosis, and cranial nerve involvement were common. Seven of 14 cases occurred in Israel, a finding suggesting the intervention of local factors. Despite antifungal and/or surgical therapy, nine patients died as a consequence of the infection days to months after diagnosis. Although a rare complication, rhinocerebral mucormycosis remains a serious threat to the kidney transplant recipient.

Adult↗

PubMed1994

Isolated central nervous system mucormycosis.

Isolated central nervous system (CNS) mucormycosis is a rare but life-threatening infection. We report a case of bilateral frontal lobe mucormycosis in a previously healthy woman. Intravenous drug use was the most likely route of infection. After treatment with surgical drainage and amphotericin B, she survived but suffered permanent neurologic deficits. We identified 29 previous reports of isolated CNS mucormycosis in the medical literature. Including our case, the patients averaged 32 years of age and most were male. Intravenous drug use appears to be the most important risk factor, present in 20 patients (67%). Treatment with amphotericin B was the only predictor of survival, reducing mortality from 92% to 41%. Isolated CNS mucormycosis should be considered in the differential diagnosis of brain abscesses, especially in intravenous drug users. Prompt initiation of therapy with amphotericin B may be life-saving.

Adult↗

PubMedDate not supplied

Indications for orbital exenteration in mucormycosis.

PURPOSE: To determine whether evidence-based standards exist regarding the indications for orbital exenteration in patients with orbital mucormycosis. METHODS: A literature review was performed of 113 articles (1943 to 2004). Factors possibly related to patient survival were analyzed. Additionally, a survey was sent to all ASOPRS Fellowship Preceptors to ascertain the frequency, outcome, treatment modalities, and indications for exenteration by these practicing physicians. RESULTS: For published cases, parameter estimates (PE 46 years, frontal sinus involvement, and fever were less likely to survive compared with patients without these conditions. Patients treated with amphotericin B (OR, 4.476) and those with diabetes (OR, 4.987) were more likely to survive compared with patients without these conditions. Exenterated patients with fever were more likely to survive compared with nonexenterated patients with fever (P=0.0468). Thirty-four ASOPRS Fellowship Preceptors received surveys; 26 (76%) completed the survey. Responses to specific survey questions showed a varied experience and indication for exenteration throughout the country. CONCLUSIONS: : Our study underscores the lack of adequate data regarding the evaluation of treatment of orbital mucormycosis. No standard of care currently exists to guide physicians on when exenteration may benefit a mucormycosis patient. Further study is needed to determine which variables indicate the extent of disease and which variables or analytic scheme might predict the progression of orbital mucormycosis with or without exenteration.

Delivery of Health Care↗

PubMed2005

Rhinocerebral mucormycosis complicated by internal carotid artery thrombosis in a pediatric patient with type 1 diabetes mellitus: a case report and review of the literature.

OBJECTIVE: To illustrate that rapid diagnosis and aggressive treatment of rhinocerebral mucormycosis with internal carotid artery occlusion in a pediatric patient can prevent mortality and significant morbidity. RESEARCH DESIGNS AND METHODS: Rhinocerebral mucormycosis infrequently occurs in the pediatric population, and when it involves thrombosis of an internal carotid artery, it has been almost uniformly fatal. We present an 8-yr-old girl with type 1 diabetes mellitus who has survived such an infection for 2 yr, and who has minimal residual morbidity. We believe she is the youngest patient to survive rhinocerebral mucormycosis complicated by internal carotid artery and cavernous sinus thromboses. She has survived with an intensive regimen including aggressive surgical debridement, amphotericin B, rigorous glucose control, hyperbaric oxygen therapy, interferon-gamma, posaconazole (an experimental antifungal), and granulocyte-macrophage colony-stimulating factor. CONCLUSIONS: This article illustrates the importance of prompt recognition and aggressive treatment of fungal infection in patients with diabetes. Additionally, it emphasizes that such treatment can have an excellent outcome, and mortality and significant morbidity can be avoided. Finally, we provide a review of the literature regarding mucormycosis infections and treatment options.

Carotid Artery Thrombosis↗

PubMed2004

Molecular polymerase chain reaction diagnosis of pulmonary mucormycosis caused by Cunninghamella bertholletiae.

OBJECTIVE: Mucormycosis is an uncommon but frequently fatal infection caused by strains of mucorales in immunocompromised hosts. In this study, we report a case of pulmonary mucormycosis associated with acute lymphocytic leukaemia caused by Cunninghamella bertholletiae, a rare pathogen. METHODOLOGY: Retrospective analysis of the stored serum, sputum, and necropsy lung tissue samples from this patient, enabled subspecies identification by means of panfungal polymerase chain reaction (PCR), direct DNA sequencing of the PCR products, and homology search with nucleotide basic local alignment search tool. RESULTS: The development of a reliable diagnostic blood test for angio-invasive fungal infections such as mucormycosis is desirable, because the sensitivity of culture for these fungi is extremely low. CONCLUSION: Panfungal PCR on serial serum samples might be useful for the diagnosis of pulmonary mucormycosis.

Cunninghamella↗

PubMed1999

Primary cutaneous mucormycosis: a diagnosis to consider.

Primary cutaneous mucormycosis is a deep fungal infection, mainly seen in diabetics and immunocompromised subjects. Rapid diagnosis and therapy are necessary to avoid fatal outcome. We describe the complete histopathological and microbiological studies of primary cutaneous mucormycosis in a 74-year-old man with several risk factors, such as chronic obstructive pulmonary disease, respiratory acidosis, hemolytic anemia, myelodysplastic syndrome and iatrogenic diabetes, due to corticosteroid therapy. He developed two cutaneous necrotic scars on his left leg. Mucormycosis was suspected and specimens from surgical débridement were histopathologically and microbiologically studied confirming the clinical diagnosis. Amphotericin B was given topically and intravenously resulting in complete healing of the ulcer. Risk factors and microbiological studies are compared with those in the current literature. It is necessary in certain cases to suspect mucormycosis infections in diabetics, immunocompromised subjects and even in healthy individuals. Rapid diagnosis and treatment are important, but they should be based on complete histopathological and microbiological studies, to establish the genus of the causal agent.

Aged↗

PubMed2005

Novel perspectives on mucormycosis: pathophysiology, presentation, and management.

Mucormycosis is a life-threatening fungal infection that occurs in immunocompromised patients. These infections are becoming increasingly common, yet survival remains very poor. A greater understanding of the pathogenesis of the disease may lead to future therapies. For example, it is now clear that iron metabolism plays a central role in regulating mucormycosis infections and that deferoxamine predisposes patients to mucormycosis by inappropriately supplying the fungus with iron. These findings raise the possibility that iron chelator therapy may be useful to treat the infection as long as the chelator does not inappropriately supply the fungus with iron. Recent data support the concept that high-dose liposomal amphotericin is the preferred monotherapy for mucormycosis. However, several novel therapeutic strategies are available. These options include combination therapy using lipid-based amphotericin with an echinocandin or with an azole (largely itraconazole or posaconazole) or with all three. The underlying principles of therapy for this disease remain rapid diagnosis, reversal of underlying predisposition, and urgent surgical debridement.

Antifungal Agents↗

PubMed1992

Pseudoaneurysm of pulmonary artery in mucormycosis. Radiographic characteristics and management.

Pulmonary mucormycosis is a rare and almost invariably fatal complication that can occur in the context of severe deficits in host defenses. Antemortem diagnosis is difficult and requires a high index of suspicion together with invasive diagnostic techniques. Mucor species exhibit a pronounced affinity to invade vessels; mucormycosis involving the pulmonary vasculature has rarely been documented antemortem, and survival in this context has been rare. In this report, we describe a patient with chronic renal failure and systemic lupus erythematosus who developed extensive invasion of the left main pulmonary artery by mucormycosis. Chest computed tomographic (CT) scans and pulmonary arteriogram demonstrated a massive pseudoaneurysm of the left pulmonary artery; these radiographic findings have not previously been described in mucormycosis. Aggressive combination therapy, employing preoperative amphotericin B (AmB) followed by surgical resection (pneumonectomy) and a full course of AmB, was curative. This favorable outcome supports the role of surgery as adjunctive therapy, and it underscores the need for early diagnosis and aggressive treatment.

Adult↗

PubMed1984

Specific susceptibility to mucormycosis in murine diabetes and bronchoalveolar macrophage defense against Rhizopus.

To assess the influence of diabetes mellitus in predisposing to pulmonary mucormycosis, a murine model of streptozotocin-induced diabetes was used. Intranasal inoculation of Rhizopus oryzae into diabetic mice resulted in mucormycotic infection with histopathology resembling pulmonary mucormycosis observed in humans. There was no mortality nor infection in inoculated normal mice. Diabetic mice had fatal infections caused by R. oryzae but significantly reduced mortality following inoculation with Aspergillus fumigatus. These findings reflect the specific enhanced susceptibility to mucormycosis observed in human diabetics. Normal bronchoalveolar macrophages formed part of an efficient defense against R. oryzae by inhibiting germination, the critical step in the conversion of R. oryzae to its tissue invasive phase. Bronchoalveolar macrophages inhibited spore germination in vitro and appeared to help prevent germination in vivo. In contrast, spore germination occurred in diabetic mice following intranasal inoculation. Diabetic bronchoalveolar macrophages had a decreased ability to attach to hyphae. In diabetic mice, bronchoalveolar macrophages could damage spores or hyphae of R. oryzae, but serum factors appeared to both promote spore germination and impair attachment of macrophages to spores. This murine model of diabetes mellitus provides an opportunity for evaluation of the relative importance of cell and serum-mediated host factors in the pathogenesis of mucormycosis.

Acid Phosphatase↗