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PubMed2006

[Rhinocerebral mucormycosis: A case report].

Mucormycosis is a rare and acute fungal infection which is frequently lethal, usually observed in non-controlled diabetic patients. The infection usually begins in the nose but it can invade the lung, the digestive tract, and the skin. Rhinocerebral mucormycosis accounts for 40 to 49% of mucormycosis cases. We report the case of a 53-year-old diabetic man, with chronic renal failure, presenting with rhinocerebral mucormycosis. Our patient was treated by an association of amphotericin B and surgical debridement. This observation provides on opportunity to recall clinical, histopathological, and therapeutic aspects of rhinocerebral mucormycosis.

Brain Diseases↗
PubMed1994

Mucormycosis (zygomycosis): is there news for the clinician?

New data on mucormycosis (zygomycosis), relevant to the clinician, are analysed. Several clinical associations of this fungal infection have recently emerged: the association with therapy with the metal chelator desferrioxamine (mainly in dialysis patients), the rarity of mucormycosis in AIDS patients and its sporadic occurrence after bone marrow transplantation. New imaging techniques should be used serially in the case of rhinocerebral mucormycosis in order to evaluate the extent of the lesions and to guide potential surgery. Surgical debridement significantly improves the prognosis of rhinocerebral mucormycosis. Finally, results of the use of liposomal amphotericin B in a few patients with rhinocerebral mucormycosis are reviewed.

AIDS-Related Opportunistic Infections↗
PubMed2006

[Rhinocerebral mucormycosis in four diabetics].

UNLABELLED: Mucormycosis is caused by a zygomycetes fungus in a vascular location. This fungus is a saprophytic organism which can become pathogenic in specific conditions, particularly in patients with diabetes mellitus. A rhinocerebral localization is common, leading to often fatal devastating sinusitis. Positive diagnosis requires histological proof with characteristic hyphal tissue invasion. Frozen section is essential for diagnosis and management of rhinocerebral mucormycosis. MATERIAL AND METHODS: We report four cases of rhinocerebral mucormycosis in diabetic patients, two men and two women, mean age 51 years. RESULTS: Histological examination showed characteristic hyphae in a vascular localization. Treatment was systemic antifungal therapy with amphotericin B and debridement of necrotic tissue. Three patients recovered completely. One died. CONCLUSION: Rhinocerebral mucormycosis is a rare fungal infection with very poor prognosis. The aim of this study was to report the clinical and pathological features of rhinocerebral mucormycosis and to evaluate the contribution of frozen section for diagnosis and management.

Adult↗
PubMed2003

Mohs micrographic surgery as an alternative treatment method for cutaneous mucormycosis.

BACKGROUND: Mucormycosis is an invasive fungal disease that most commonly occurs in immunocompromised patients. Early angioinvasion and dissemination can lead to the rapid demise of the patient. The growing number of organ transplant patients on pharmacologic immunosuppression has increased the risk for this opportunistic mycosis. Traditional therapy has included aggressive debridement and resection as well as antifungal medications. OBJECTIVE: To demonstrate that the margin control and tissue-sparing technique of Mohs micrographic surgery can effectively eradicate mucormycosis infection and decrease morbidity. METHODS: Case presentation of a 64-year-old transplant patient presenting with biopsy-proven cutaneous mucormycosis treated with Mohs micrographic surgery. Margin control was confirmed by a rapid Gomori methenamine silver stain. RESULTS: There has been no recurrence at 1-year follow-up with full preservation of extremity function. CONCLUSION: The use of the Mohs technique combined with rapid Gomori methenamine silver staining for mucormycosis can be an effective tissue-sparing method for local control of this fungal infection. Mohs micrographic surgery should be considered for the cutaneous manifestations of mucormycosis.

Dermatomycoses↗
PubMedDate not supplied

Adjunctive hyperbaric oxygen for treatment of rhinocerebral mucormycosis.

The therapy of rhinocerebral mucormycosis includes aggressive surgical debridement, administration of high-dose amphotericin B, and control of underlying predisposing conditions, especially diabetes and immunosuppression or immunodeficiency. Hyperbaric oxygen suppresses fungal growth in vitro and has theoretical value in treating mucormycosis because it reduces the tissue hypoxia and acidosis that accompany vascular invasion by the fungus. In a retrospective review of patients at Duke University Medical Center with rhinocerebral mucormycosis, six patients were treated with hyperbaric oxygen and seven cases (involving six patients) were treated without hyperbaric oxygen. All patients received surgical debridement and amphotericin B. Two of six patients receiving hyperbaric oxygen therapy died, and four of seven patients not receiving hyperbaric oxygen therapy died. Adverse effects from hyperbaric oxygen were minimal. Because mucormycosis occurs infrequently, this retrospective review involved a small number of patients. Despite this limitation, adjunctive hyperbaric oxygen appears to be a promising clinical modality for the treatment of rhinocerebral mucormycosis and warrants further investigation.

Adolescent↗
PubMed2006

Deferiprone iron chelation as a novel therapy for experimental mucormycosis.

OBJECTIVES: Patients treated with the iron chelator deferoxamine are known to be more susceptible to mucormycosis. However, while deferoxamine is an iron chelator from the perspective of the human host, deferoxamine actually serves as a siderophore, delivering free iron to Rhizopus oryzae, the major cause of mucormycosis. Other iron chelators, including deferiprone, which do not deliver iron to R. oryzae have been described. We therefore sought to determine whether iron-chelation therapy with deferiprone would effectively treat mucormycosis. METHODS: In vitro MIC and minimum fungicidal concentration (MFC) of the iron chelator, deferiprone, for R. oryzae were determined by microdilution assay. In addition, we compared the efficacy of deferiprone with that of liposomal amphotericin B (LAmB) in treating mucormycosis in diabetic ketoacidotic mice. RESULTS: Deferiprone demonstrated static activity against R. oryzae at 24 h, but showed cidality at 48 h of incubation. Deferiprone was as effective as LAmB at improving survival and decreasing brain fungal burden, and both drugs were more effective than placebo in non-iron-overloaded animals. Administration of free iron with deferiprone reversed protection, confirming that the mechanism of protection was iron chelation. CONCLUSIONS: Iron chelation is a promising, novel therapeutic strategy for refractory mucormycosis infections. Further studies are warranted to evaluate combination antifungal/iron chelation therapy and to evaluate the efficacy of other iron-chelating agents.

Amphotericin B↗
PubMedDate not supplied

Localized mucormycosis following intramuscular corticosteroid. Case report and review of the literature.

A case of subcutaneous localized mucormycosis infection which developed following intramuscular (IM) injection of corticosteroid in a patient with leukemia is presented. Aggressive treatment, which included wide local excision, systemic amphotericin-B, and chemotherapy for the leukemia, resulted in eradication of the infection and complete healing of the wound. A review of the literature revealed nine other patients with the localized subcutaneous form of mucormycosis (excluding patients with burns and rhinocerebral types) and six of those nine patients also survived the infection. It is possibly the mildness of the underlying predisposing factors that allows some of these patients to contain the infection at a single site. It is apparent from review of the literature that in subcutaneous localized forms of mucormycosis, the outcome has been generally good. This contrasts sharply with other clinical forms of mucormycosis infections where the underlying predisposing factors are usually severe and any kind of therapeutic approach has been almost always futile. Subcutaneous mucormycosis infection differs clinically and histopathologically from subcutaneous localized entomophthoromycosis which is seen predominantly in tropical countries. An attempt is made to clarify the terminology of these interesting fungi in language that is taxonomically up-to-date and still useful to clinicians.

Adult↗
PubMed2002

Primary cutaneous mucormycosis: guide to surgical management.

Mucormycosis is the most acute, fulminate, and fatal of all fungal infections in humans. It presents most frequently in immunocompromised patients, but can occur in healthy patients in the presence of often-insignificant trauma. Surgical management of primary cutaneous mucormycosis is almost always required. Case reports of surgical treatment for primary cutaneous mucormycosis are reported in the literature; however, the extent of debridement required for cure is unclear and no uniform plan of treatment has been suggested. To date, no clinical guidelines exist to assist the clinician in the surgical management of this disease. This article reviews the literature, reports on two clinical cases, and submits clinical guidelines designed to assist the clinician in the surgical management of primary cutaneous mucormycosis. Because of the infrequent and potentially fatal nature of the diagnosis, a high index of suspicion and a low threshold for wound biopsy must be maintained. Wound cultures are grossly inadequate and should not be relied on for a false sense of security. It is recommended that, for the early diagnosis of cutaneous mucormycosis, chemotherapy and surgical debridement of grossly necrotic tissue be performed at the earliest possible time. The debrided wound is monitored for the resolution of surrounding erythema and induration before definitive reconstruction. In the case of delayed diagnosis and/or advanced or rapidly progressive disease, surgical debridement of all involved tissue, in addition to chemotherapy, is warranted.

Debridement↗
PubMed1999

Long-term survival of a patient with invasive cranial base rhinocerebral mucormycosis treated with combined endovascular, surgical, and medical therapies: case report.

OBJECTIVE: Rhinocerebral mucormycosis is a clinical syndrome resulting from an opportunistic infection caused by a fungus of the order Mucorales. The prognosis of rhinocerebral mucormycosis, once considered uniformly fatal, remains poor. Even with early diagnosis and aggressive surgical and medical therapy, the mortality rate is high. We present a patient with rhinocerebral mucormycosis involving the paranasal sinuses and cranial base who experienced long-term survival after multimodality treatment. Clinical characteristics of the disease are discussed, and the literature is reviewed. CLINICAL PRESENTATION: A 24-year-old diabetic man presented with invasive rhinocerebral mucormycosis involving the paranasal sinuses, right middle fossa, and right cavernous sinus. INTERVENTION: The patient underwent endovascular sacrifice of the involved carotid artery and radical resection of the cranial base, including exenteration of the cavernous sinus. Reconstruction with a local muscle flap was performed. He continued to receive intravenous and intrathecal administration of antibiotics. CONCLUSION: Long-term survival with invasive rhinocerebral mucormycosis is rare, but possible, with aggressive multimodality treatment, including carotid sacrifice for en bloc resection of the pathology, when indicated.

Adult↗
PubMed2004

Mucormycosis and entomophthoramycosis: a review of the clinical manifestations, diagnosis and treatment.

The class Zygomycetes is divided into two orders, Mucorales and Entomophthorales. These two orders produce dramatically different infections. Genera from the order Mucorales (Rhizopus, Mucor, Rhizomucor, Absidia, Apophysomyces, Cunninghamella and Saksenaea) cause an angioinvasive infection called mucormycosis. Mucormycosis presents with rhino-orbito-cerebral, pulmonary, disseminated, cutaneous, or gastrointestinal involvement. Immunocompromising states such as haematological malignancy, bone marrow or peripheral blood stem cell transplantation, neutropenia, solid organ transplantation, diabetes mellitus with or without ketoacidosis, corticosteroids, and deferoxamine therapy for iron overload predispose patients to infection. Mucormycosis in immunocompetent hosts is rare, and is often related to trauma. Mortality rates can approach 100% depending on the patient's underlying disease and form of mucormycosis. Early diagnosis, along with treatment of the underlying medical condition, surgery, and an amphotericin B product are needed for a successful outcome. Genera from the order Entomophthorales produce a chronic subcutaneous infection called entomophthoramycosis in immunocompetent patients. This infection occurs in tropical and subtropical climates. The genus Basidiobolus typically produces a chronic subcutaneous infection of the thigh, buttock, and/or trunk. Rarely, it has been reported to involve the gastrointestinal tract. The genus Conidiobolus causes a chronic infection of the nasal submucosa and subcutaneous tissue of the nose and face. This paper will review the clinical manifestations, diagnosis and treatment of mucormycosis and entomophthoramycosis.

Humans↗
PubMed1995

Mucormycosis caused by Rhizopus microsporus var. microsporus: cellulitis in the leg of a diabetic patient cured by amputation.

Mucormycosis accompanied the development of bacterial infection in the leg of a diabetic African-American man. Local injury, diabetic ketoacidosis, renal insufficiency, and antimicrobial therapy were factors that contributed to the pathogenesis of the mucormycosis. The cellulitis was caused in part by Rhizopus microsporus var. microsporus and was cured by amputation. We report this unusual case of mucormycosis to emphasize the value of fungal identification, to illustrate a dramatic and successful clinical result, and to draw attention to an apparent role for bacterial infection and its treatment in the pathogenesis of mucormycosis. It is the third case report of mucormycosis in a human in which R. microsporus var. microsporus was definitively identified as the etiologic agent.

Adult↗
PubMed2002

Palate ulcer due to mucormycosis.

Mucormycosis is caused by fungi of the order Mucorales and is one of the most rapidly fatal fungal infections known to man. Rhinocerebral mucormycosis is the most common type and its extension to the orbit and brain is quite usual. Location of mucormycosis on the palate is a rare and late occurrence. A case of deep hard palate ulcer due to sinonasal mucormycosis in a 79-year-old man is reported. He was successfully treated with a combination of surgical debridement and systemic liposomal amphotericin B administration for six weeks. By presenting this case report we would like to point out that mucormycosis should be included in the differential diagnosis of hard palate ulcers.

Aged↗
PubMed1992

Intracranial complications of mucormycosis: an experimental model and clinical review.

The clinical course of patients with mucormycosis of the paranasal sinuses can be unpredictable and is often determined by intrinsic host factors. The degree to and mechanism(s) by which these factors influence a patient's ability to survive the disease are poorly understood. Extensions to orbital and intracranial structures occur in some patients with paranasal sinus mucormycosis despite aggressive treatment. Controversies persist over adequate antifungal regimen, the precise role of hyperbaric oxygenation, and the appropriate extent of surgical debridement. We have developed an alloxan-induced immunocompromised murine model of mucormycosis in mice. Deferoxamine iron chelation produced rhinocerebral mucormycosis in these animals when challenged intraethmoidally with Rhizopus spores. The implications of our experimental studies in the content of our clinical experience in managing patients with intracranial extensions of paranasal sinus mucormycosis are discussed.

Adult↗
PubMed1986

Clinical spectrum of pulmonary mucormycosis.

Pulmonary mucormycosis is an uncommon, but important, opportunistic fungal pneumonia which is often diagnosed post-mortem. This review emphasizes clinical and pathologic characteristics of pulmonary mucormycosis that differentiate this infection from other fungal pneumonias. The most common clinical presentation of pulmonary mucormycosis is a rapidly progressive pneumonia with diffuse infiltrates on chest radiographic examination of a patient with an underlying hematologic malignancy treated with immunosuppressive drugs. Other immunocompromised hosts at risk for pulmonary mucormycosis include patients with diabetes mellitus who may develop a distinctive endobronchial form of this disease. Early consideration of this diagnosis, along with aggressive diagnostic evaluation, are critical to effective therapy and patient survival. While treatment with amphotericin B is the mainstay of therapy for pulmonary mucormycosis, diabetics with endobronchial disease may benefit from early, aggressive surgical resection of the involved lung tissue.

Adult↗
PubMed2005

Mucormycosis in immunocompetent individuals: an increasing trend.

OBJECTIVE: Mucormycosis is a fatal infection of the immunocompromised individual. It is unusual to affect healthy individuals. We report eight such cases of infection caused by this emerging fungal pathogen in healthy patients. Of the eight cases, three were infected with Apophysomyces elegans, again an unusual pathogen causing mucormycosis. STUDY DESIGN: Retrospective case review conducted at a tertiary referral center. METHODS: From 1999 to 2003, eight cases of mucormycosis were managed in otherwise healthy patients. Seven of them were treated with surgery. Clinical presentation, imaging studies, mycologic findings, operative findings at surgery, and postoperative results were evaluated. A review of the literature pertaining to mucormycosis infecting otherwise healthy patients and A. elegans infecting otherwise healthy patients in the nose and paranasal sinuses also was done. RESULTS: Of the eight cases, three were infected with A. elegans, with no history of trauma or any invasive procedure. Seven patients underwent surgical treatment. Histopathologic examination showed broad, sparsely aseptate, thin-walled hyphae and angioinvasion with thrombosis. CONCLUSION: Mucormycosis must be considered in the differential diagnosis of any severe acute headache, sinusitis, or orbital cellulites, not only in immunocompromised patients but also in the absence of any underlying disease. Successful treatment requires tissue débridement and injection of amphotericin B.

Adult↗
PubMed2002

[Rhinocerebral mucormycosis: report of a rare case in the head-neck and chest area].

Mucormycosis is a severe fungal disease. It can present in two clinical forms: localized or disseminated. The most common, often lethal, form is localized rhinocerebral mucormycosis and is associated with diabetes, debilitation and immunologic deficiencies. The primary location is generally found in the nasal cavity, paranasal sinuses or hard palate. The infective process can extend into the orbita, the cavernosus sinus, the pterygium-palatine and infratemporal fossae with generation of mycotic emboli that can plug the cerebral arteries and, in certain circumstances, can lead to death if it goes untreated. This work describes a clinical case of a 76-years-old patient with a previous history of alcoholism and glucose intolerance, affected by rhinocerebral mucormycosis with extension to the lungs. A general medicine division sent the patient for observation by a specialist since, at the time of hospitalization, objective testing showed signs of an ulcerating lesion of the alveolar edge of the left hemipalate associated with a bilateral pulmonary neoformation suspected to be neoplastic. The patient underwent pulmonary and maxillo-facial surgical resection and medical therapy. The authors discuss the appropriateness of diagnosis and treatment procedures in cases of mucormycosis in association with other pathologies, that can complicate the clinical picture, delaying diagnosis and treatment. The authors point out the need to perform in a short time an adequate medical and surgical treatment of mucormycosis, because of the risk of intracranial extension, leading to an increased mortality, even as high as 80%. The longer it takes to reach a diagnosis the more radical the surgical treatment will need to be.

Aged↗
PubMed2003

Rhinocerebral mucormycosis in Taiwan.

To understand the demographic as well as clinical characteristics, and outcomes of patients with rhinocerebral mucormycosis in Taiwan, we retrospectively analyzed patients with this disease admitted to Chang Gung Memorial Hospital-Kaohsiung from 1988 through 2000. The 21 patients included 8 men (28%) and 13 women (62%). The median age was 60 years (range, 34-82 years). Twenty patients (95%) had underlying diabetes mellitus. The most common clinical feature at admission was ocular lesions, followed by headache, nostril lesions, and consciousness disturbance. Fifteen (94%) of 16 patients who received combined surgical debridement and therapy with amphotericin B survived, while only 1 (20%) of the 5 patients who received amphotericin B alone survived (p=0.004). The diagnosis of rhinocerebral mucormycosis was delayed in 4 patients (19%). Of the 16 patients who survived, 1 (6%) had delayed diagnosis, while of the 5 patients who died, 3 (60%) had delayed diagnoses (p=0.028). This series disclosed a higher proportion of patients with rhinocerebral mucormycosis in Taiwan had underlying diabetes mellitus, and ocular lesions were more frequent than nostril lesions at the time of admission. These results highlight the importance of the timely initiation of a combination of aggressive surgical debridement and treatment with amphotericin B in patients with rhinocerebral mucormycosis. Considering the high rate of delayed diagnosis, improved clinician's awareness of mucormycosis is extremely important and is in urgent need in Taiwan.

Adult↗
PubMed2004

[Mucormycosis].

Over recent years the clinical importance of invasive fungal infections has significantly increased. Mucormycosis refers to a rare, life threatening, opportunistic disease caused by filamentous fungi of the order Mucorales. Most frequently mucormycosis occurs in patients with ketoacidotic diabetes and in neutropenic patients with acute leukaemia. This study of fifteen patients was conducted to evaluate the clinical characteristics of invasive mucormycosis, and goal was also to propose for an early diagnosis and management as survival can only be improved by aggressive surgery and antifungal therapy. In a retrospective analysis of the biopsy and autopsy records of the Department of Pathology at Szent László Hospital between 1992-2003 were evaluated fifteen histologically documented cases of mucormycosis. The underlying diseases were hematological malignancy with neutropenia in ten cases, chronic leukaemia with neutropenia, chronic leukaemia with latent diabetes mellitus and diabetes mellitus one case each. Three cases did not belong to any typical risk group. An in vivo diagnosis was made only in four patients with a rhinocerebral manifestation. Radical necrectomy and long-term antifungal therapy cured two of them. Two patients died of a generalised infection despite therapy. None of the systemic infections could be cured. The diagnosis was made post mortem in eleven patients. Selected clinical forms of mucormycosis are illustrated with short case reports.

Adolescent↗