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Localized muscular mucormycosis in a child with acute leukemia.

Mucormycosis is a rare fungal infection of childhood, occurring mainly in patients with chronic illnesses such as diabetes and malignancies. The fungus seldom grows in culture and confirmation of the diagnosis depends on histologic examination of infected tissues. To date, the reported natural history of the disease has been rapid progression and a fatal outcome. Therefore, the importance of early diagnosis by tissue biopsy and early treatment with surgical debridement and systemic antifungal therapy cannot be overemphasized. The pulmonary system is the most common site for mucormycosis in patients with leukemia. We report what we believe to be the first successfully treated case of isolated muscular mucormycosis occurring in a child with biphenotypic acute leukemia. The diagnosis was made promptly by tissue examination at the time of surgical debridement. The patient was also given systemic amphotericin-B therapy.

Acute Disease↗

Arterial occlusion and progressive gangrene caused by mucormycosis in a patient with burns.

Ischemic necrosis of the upper extremities caused by invasive mucormycosis developed in a patient with soil contamination of severe burn wounds. An arteriogram of the arm showed complete obstruction of blood flow in the forearm. Histologic specimens showed nonseptate branching hyphae obliterating the arterial lumens. Cutaneous mucormycosis affects patients who are immunocompromised, including victims of multiple trauma and burns. This case represents a previously undescribed clinical presentation in a patient with major burns. Because of its lethal nature, mucormycosis in a patient with burns must be treated with aggressive surgical debridement, including amputation, and with parentral amphotericin B at the earliest sign of cutaneous presence.

Adult↗

Rhinocerebral mucormycosis in patients with burns: case report and review of the literature.

Mucormycosis is an opportunistic infection most commonly occurring in patients with impaired host defenses or diabetes mellitus. In patients with burns the rhinocerebral form is rare, and mucormycotic infections more commonly involve the cutaneous burn wound. Both forms are associated with a high mortality rate that increases with delays in treatment. The initial management of these types of infections includes vigorous glucose control, correction of acidosis, and the administration of systemic antifungal agents such as amphotericin B. The rhinocerebral form of mucormycosis is extremely virulent and may warrant the use of interstitial and intraventricular antifungal therapy. Despite these measures, the mainstay of treatment for both forms of mucormycosis is the extensive surgical débridement of all infected and necrotic tissue.

Antifungal Agents↗

Case report. Nasopharyngeal mucormycosis.

Successful management of nasopharyngeal mucormycosis in a 58-year-old leukemia patient is reported. High index of suspicion, localizing the disease to the nasopharynx, early diagnosis by biopsy, control of underlying debilitating leukemia, and aggressive therapeutic approach all contributed to the successful outcome. Review of the literature indicates this to be the first reported case of successful outcome of mucormycosis in a patient with acute myelogenous leukemia. Application and findings of computed tomography for evaluation of mucormycosis are discussed.

Female↗

Mucormycosis of the central airways: CT findings in three patients.

Computed tomographic (CT) findings are described in three diabetic patients with central airways mucormycosis. The CT findings of the tracheobronchial mucormycosis include enhancing areas of mural thickening (n = 3), luminal narrowing (n = 3), intramural air (n = 3), low-attenuation nonenhancing bronchial wall thickening (n = 2), and bronchonodal fistula formation (n = 1). These CT features in a diabetic patient should raise a high index of suspicion for tracheobronchial mucormycosis, particularly when typical radiographic features of pulmonary tuberculosis are absent.

Aged↗

Chronic rhinocerebral mucormycosis.

Rhinocerebral mucormycosis is a well-described fulminant fungal infection that typically presents acutely in patients with diabetic ketoacidosis or immunosuppression. Chronic presentations of rhinocerebral mucormycosis have also been described. In the chronic infection, the disease course is indolent and slowly progressive, often occurring over weeks to months. The authors report 2 cases of chronic rhinocerebral mucormycosis (CRM) treated at their institution and review 16 other cases reported in the English-language literature. In these cases, the median time from symptom onset to diagnosis was 7 months. The most common presenting features of CRM are ophthalmologic and include ptosis, proptosis, visual loss, and ophthalmoplegia. CRM occurs predominantly in patients with diabetes and ketoacidosis. The incidence of internal carotid artery and cavernous sinus thrombosis is higher in CRM patients than in those with the acute disease, although the overall survival rate for CRM patients is 83%. CRM is clinically distinct from chronic Entomophthorales infection.

Aged↗

Rhinocerebral mucormycosis: evolution of the disease and treatment options.

Rhinocerebral mucormycosis is recognized as a potentially aggressive and commonly fatal fungal infection. The classic presentation is involvement of nasal mucosa with invasion of the paranasal sinuses and orbit. Mucormycosis is most commonly seen in association with diabetic ketoacidosis, but disease demographics have changed with the onset of AIDS and the advent of powerful immunosuppressive drugs. Treatment includes aggressive debridement, systemic antifungal therapy, and control of underlying comorbid factors. Although surgical intervention remains essential, advances in medical therapy have permitted a more limited surgical approach to minimize functional loss without compromising survival. We present the UCLA experience with rhinocerebral mucormycosis from 1955 to 1995, with emphasis on the evolution of disease presentation and alternative treatment options.

AIDS-Related Opportunistic Infections↗

Treatment of rhinocerebral mucormycosis with intravenous interstitial, and cerebrospinal fluid administration of amphotericin B: case report.

IMPORTANCE: Rhinocerebral mucormycosis is extremely difficult to treat. Approximately 70% of patients are poorly controlled diabetics, and many of the remainder are immunocompromised as a consequence of cytotoxic drugs, burn injuries, or end-stage renal disease. Despite standard treatment consisting of surgical debridement and the intravenous administration of amphotericin B, rhinocerebral mucormycosis is usually a fatal disease. CLINICAL PRESENTATION: We describe the case of a 16-year-old male patient with juvenile onset diabetes mellitus who presented with fever, right-sided hemiparesis, and dysarthria. Axial view computed tomography revealed abscess formation in the left basal ganglia and frontal lobe, which was proven by stereotactic biopsy to contain Rhizopus oryzae. INTERVENTION: Intravenous administration of amphotericin B (30-280 mg/dose) was begun on the day of admission. On hospital Day 20, after the occurrence of frank abscess formation, the lesion was aggressively debrided. Despite these therapies, there was neurological deterioration characterized by the development of hemiplegia and aphasia. Sequential computed tomographic scans enhanced with contrast medium demonstrated progressively enlarging lesions. Ommaya reservoirs were placed into the abscess cavity and the frontal horn of the contralateral lateral ventricle. The patient was then treated with intracavitary/interstitial injections of amphotericin B during the course of 80 days and three doses of intraventricular amphotericin B. Clinical and radiographic improvement was achieved after treatment. Two years after the initial diagnosis, magnetic resonance imaging of the brain showed no evidence of disease and an examination revealed a neurologically intact and fully functional patient. CONCLUSION: We conclude that with an infection as morbid as rhinocerebral mucormycosis, it is advisable to use surgical debridement and all available routes for delivering amphotericin B to infected cerebral parenchyma, which include intravenous, intracavitary/interstitial, and cerebrospinal fluid perfusion pathways.

Adolescent↗

Nonfatal gastric mucormycosis in a renal transplant recipient.

Mucormycosis historically has caused substantial morbidity with high mortality in renal transplant patients with disseminated and/or rhinocerebral infection and in patients with gastrointestinal illness regardless of predisposing conditions. We report the first successful treatment of gastric mucormycosis in a renal transplant recipient and review presumed pathogenic mechanisms of mucormycosis in renal transplant recipients as well as historical data.

Amphotericin B↗

Early onset of pulmonary mucormycosis with pulmonary vein thrombosis in a heart transplant recipient.

Pulmonary infections are major complications in heart transplantation. Progress in antimicrobial chemotherapy has switched the clinical spectrum to an increased incidence of fungal pathogens, such as Candida and Aspergillus species. Mucormycosis is a rare opportunistic infection with high mortality in solid-organ transplant recipients usually ensuing several months after transplantation. We describe a 45-year-old patient with pulmonary mucormycosis manifestion 5 days after heart transplantation. The infection resulted in pulmonary vein thrombosis followed by hemorrhagic infarction. Despite antifungal treatment and surgical resection, the patient died on day 14 after transplantation. Antemortem diagnostic procedures were negative; autopsy confirmed the presence of Rhizopus oryzae invading blood vessels. We conclude that physicians must be aware of mucormycosis even within one week after heart transplantation--which has not been described so far. Invasive diagnostic workup is mandatory in case of suspicion; amphotericin B and, in selected cases, surgical resection are the mainstays of therapy.

Heart Transplantation↗

Rhinocerebral mucormycosis in a boy with recurrent acute lymphoblastic leukemia: long-term survival with systemic antifungal treatment.

Rhinocerebral mucormycosis is rare in hematologic malignancies and usually leads to death within weeks. In contrast, chronic rhinocerebral mucormycosis takes a slowly progressive course and has not been reported in hematologic malignancies in children so far. The authors report the long-term survival of a boy with rhinocerebral mucormycosis in a relapse of acute lymphoblastic leukemia after allogeneic cord blood transplantation. The disease started acutely but took a chronic course thereafter. No surgical debridement was performed because of extensive involvement of the sinuses, orbits, and cerebrum. His long-term survival of 15 months is attributed to the long-range administration of liposomal amphotericin B, early neutrophil recovery, and slow progression of the relapsing acute lymphoblastic leukemia.

Amphotericin B↗

Successful treatment of disseminated mucormycosis with a combination of liposomal amphotericin B and posaconazole in a patient with acute myeloid leukaemia.

The combination of resection of infected tissue and antifungal therapy is the treatment of choice in mucormycosis. In disseminated mucormycosis, where surgery is impossible, the mortality is almost 90%. We report the first case of disseminated mucormycosis that was cured with a combination therapy of liposomal amphotericin B and posaconazole without surgical intervention.

Amphotericin B↗

Intestinal mucormycosis in a hemodialysis patient treated with desferrioxamine.

A case of fatal intestinal mucormycosis in a 57-year-old female hemodialysis patient who had received desferrioxamine (DFO) for aluminum overload is reported. The focus of fungal infection was not found until the intestine had been resected surgically. DFO is being used with increased frequency in dialysis patients to treat aluminum or iron overload. Recently a possible link between DFO therapy and mucormycosis has been suggested. In the management of hemodialysis patients, the potential risk of mucormycosis with DFO therapy should be considered.

Aluminum↗

Combination antifungal therapy in treatment of murine pulmonary mucormycosis: roles of quinolones and azoles.

Amphotericin B is the only recognized antifungal used in the treatment of mucormycosis. In this study, we evaluated various combinations of amphotericin B, fluconazole, and trovafloxacin or ciprofloxacin in the treatment of murine pulmonary mucormycosis. The combination of fluconazole and a quinolone has a marked effect on the outcome of murine pulmonary mucormycosis. Even though we did not optimize therapy with the drugs, these experiments suggest that azoles, especially fluconazole, in combination with either trovafloxacin or ciprofloxacin were effective in the treatment of this aggressive mycosis in the mouse model.

4-Quinolones↗

Craniofacial mucormycosis: assessment with CT.

Computed tomographic (CT) scans of ten patients with rhinocerebral mucormycosis were reviewed. Early paranasal sinus involvement appeared as mucosal thickening on CT scans, usually without air/fluid levels. Recognition as mucormycosis was facilitated by knowledge of the clinical setting or by identification of invasive disease. Evidence of bone destruction on CT scans was seen in only two patients, was a late finding, and usually was absent despite deep extension of disease beyond the bony confines of the paranasal sinus. Five cases had intracranial involvement, either as fungal abscess or infarction. Intracranial mucormycosis usually involved the base of the brain and cerebellum following invasion of the infratemporal fossa or orbit. Intracerebral fungal abscess appeared as low-density masses on CT scans, with variable peripheral enhancement and little surrounding vasogenic edema. Identification of a rim of spared cortex was useful in distinguishing infection from bland infarct. Serial CT scans were also useful in assessing response to hyperbaric oxygen treatment, surgery, and chemotherapy.

Adult↗

Successful treatment of massive lower gastrointestinal bleeding caused by mixed infection of cytomegalovirus and mucormycosis in a renal transplant recipient.

We describe a case of lower gastrointestinal bleeding due to mixed infection of cytomegalovirus (CMV) and mucormycosis in a renal transplant recipient. A 33-year-old male received renal transplantation and his clinical course was uneventful. On the 18th postoperative day, acute rejection was developed and this was treated with high-dose methylprednisolone and OKT3. During antirejection treatment, sudden onset massive hematochezia was developed. Emergency colonofibroscopy revealed multiple colonic ulcers and pathologic findings were consistent with mucormycosis and CMV infection. The patient was successfully treated with amphotericin B (1.0-1.5 mg/kg) and ganciclovir (62.5-125 mg/day) for 5 weeks. To our knowledge, this is the first report showing coexistence of mucormycosis and CMV in the colon ulcer base. This finding suggests that CMV infection may trigger fungal infection in the pathogenesis of colonic ulcer.

Adult↗

Pulmonary mucormycosis in the setting of chronic obstructive pulmonary disease. A case report and review of the literature.

We describe the first case of pulmonary mucormycosis occurring in a patient with chronic obstructive pulmonary disease (COPD) maintained on chronic low dose oral steroids (10 mg/day). The diagnosis was made by direct histopathological examination and culturing of infected tissue obtained by fiberoptic bronchoscopy. Pulmonary mucormycosis is caused by infection with an opportunistic fungus of the order Mucorales and is an acute, rapidly developing and often fulminant process usually occurring in immunocompromised individuals. Risk factors include neutropenia, hematologic malignancies, uncontrolled diabetes mellitus, skin burns and deferoxamine therapy in dialysis patients. This case illustrates the importance of early suspicion of mucormycosis and immediate diagnostic bronchoscopic examination in cases of rapidly progressing pulmonary infiltrates in COPD patients on low doses of corticosteroids.

Aged↗

Unexpected expansive paranasal sinus mucormycosis.

Mucormycosis infection of the paranasal sinuses is a very rare but serious condition. Patients are usually immunocompromised by systemic disease. We report an unexpected case of expansive paranasal sinus mucormycosis in an immunocompetent European treated successfully with surgical debridement and intravenous amphotericin B. The pathogenesis, diagnosis and treatment of paranasal sinus mucormycosis is discussed.

Anti-Bacterial Agents↗