Colonic basidiobolomycosis in a patient with systemic lupus erythematosus (SLE).

BACKGROUND: Basidiobolus ranarum belongs to the Entomophthorales order and the Zygomycetes class. This fungus is an environmental saprophyte that can be found in soil and rotting vegetables.Primarily restricted to tropical regions including Asia, Africa, and South America. It might cause chronic inflammatory diseases, mostly affect subcutaneous tissue. Systemic infections involving the gastrointestinal tract are extremely rare. CASE PRESENTATION: Herein, we present a 44-year-old Persian man with the past medical history of lupus erythematosus with colicky abdominal pain started from three months before admission with many vomiting episodes, and a mass on the right lower quadrant, who had been thought initially to have an abdominal malignancy. The patient had vital signs were within normal ranges. His physical examination revealed tenderness and rebound tenderness on the right lower quadrant of the abdomen. A fixed mass 10 × 10 centimeter in diameter was palpated in the same quadrant. Laboratory, radiologic, colonoscopic examination was requested. The patient underwent laparotomy which revealed a mass in the terminal ileum and ascending colon with retroperitoneal adhesion and invasion to the right ureter behind it. Pathologic examination showed basidiobolomycosis infection in the specimen. CONCLUSION: Fungal infection should be among the differential diagnoses for adults present with abdominal mass in endemic regions of the world.

Conidiobolus pachyzygosporus invasive pulmonary infection in a patient with acute myeloid leukemia: case report and review of the literature.

BACKGROUND: Conidiobolus spp. (mainly C. coronatus) are the causal agents of rhino-facial conidiobolomycosis, a limited soft tissue infection, which is essentially observed in immunocompetent individuals from tropical areas. Rare cases of invasive conidiobolomycosis due to C. coronatus or other species (C.incongruus, C.lamprauges) have been reported in immunocompromised patients. We report here the first case of invasive pulmonary fungal infection due to Conidiobolus pachyzygosporus in a Swiss patient with onco-haematologic malignancy. CASE PRESENTATION: A 71 year-old female was admitted in a Swiss hospital for induction chemotherapy of acute myeloid leukemia. A chest CT performed during the neutropenic phase identified three well-circumscribed lung lesions consistent with invasive fungal infection, along with a positive 1,3-beta-d-glucan assay in serum. A transbronchial biopsy of the lung lesions revealed large occasionally septate hyphae. A Conidiobolus spp. was detected by direct 18S rDNA in the tissue biopsy and subsequently identified at species level as C. pachyzygosporus by 28S rDNA sequencing. The infection was cured after isavuconazole therapy, recovery of the immune system and surgical resection of lung lesions. CONCLUSIONS: This is the first description of C. pachyzygosporus as human pathogen and second case report of invasive conidiobolomycosis from a European country.

Gastrointestinal basidiobolomycosis in a patient suffering from duodenal ulcer with perforation: First case report from Italy.

Gastrointestinal basidiobolomycosis (GIB), an unusual fungal infection caused by the fungus Basidiobolus ranarum, is rarely reported in the medical literature. GIB is difficult to diagnose because its clinical presentation is non-specific and has no identifiable risk factors. We report here the first case of GIB diagnosed in Italy in a patient suffering from a duodenal ulcer with perforation. The patient was successfully treated with itraconazole. The absence of non-specific signs and symptoms of GIB may delay a definitive diagnosis and treatment. A microbiological investigation should always be requested in order to reach a rapid and definitive diagnosis and to rule out other intestinal diseases.

Neonatal Acute Liver Failure Associated with Angioinvasive Hepatic Zygomycosis.

BACKGROUND: Neonatal acute liver failure (NALF) is often a fatal condition. Zygomycosis is a fungal infection that is often fatal in both adults and infants. Only a few cases of hepatic zygomycosis are reported in the literature and they are invariably associated with immunosuppression. MATERIALS AND METHODS: Post-mortem liver biopsy from a 14-day old neonate demonstrated confluent panacinar necrosis with angioinvasive zygomycosis. The limited work-up could not rule out an underlying immunodeficiency. CONCLUSION: Angioinvasive hepatic zygomycosis can present in the neonatal period as NALF.

Cutaneous basidiobolomycosis: Seven cases in southern Benin.

BACKGROUND: Cutaneous basidiobolomycosis is the most common form of entomophthoramycosis. Herein we report seven cases of cutaneous basidiobolomycosis. PATIENTS AND METHODS: A retrospective observational study was conducted at the Buruli ulcer treatment centre in Pobè and at the national teaching hospital in Cotonou from 2010 to 2015. RESULTS: Seven cases of cutaneous basidiobolomycosis were diagnosed. The mean patient age was 9.53 years. There were 4 female and 3 male patients, all from southeast Benin. Clinically, the disease presented in all cases as a hard, well-defined, subcutaneous plaque with little inflammation, and which could easily be lifted from the deep structures but remained attached to the surface structures. The overlying skin was hyperpigmented. Plaques were localized to the buttocks or thighs. All patients had inflammatory anaemia with an accelerated erythrocyte sedimentation rate (30 to 70mm over the first hour), and a low haemoglobin count (8.7 to 11.4g/dL). Blood hypereosinophilia (650 to 3784elements/mm3) was present in six of the seven subjects. Histopathology (performed for 5 of the 7 subjects) showed granulomatous lesions with foreign-body giant cells, and inflammatory cells, with occasional eosinophils surrounding fungal hyphae (Splendore-Hoeppli phenomenon). Mycological analysis revealed Basidiobolus ranarum in three cases. The patients were treated with ketoconazole (5/7) and itraconazole (2/7), with good outcomes after 10 to 24 weeks of therapy. DISCUSSION: Cutaneous basidiobolomycosis is uncommon in southern Benin, with only seven cases being diagnosed over 6 years. The diagnosis of cutaneous basidiobolomycosis is a challenge in the field in Benin due to the non-specific clinical presentation, the lack of technical resources, and the existence of numerous differential diagnoses. CONCLUSION: Cutaneous basidiobolomycosis is an uncommon fungal infection in southern Benin chiefly affecting children.

Zygomycosis Presenting as Acute Bilateral Renal Artery Thrombosis in a Healthy Young Male.

We present a case of acute renal artery thrombosis due to disseminated zygomycosis in a healthy young adult male.The diagnosis of renal artery occlusion was made on contrast-enhanced CT (CECT) and confirmation of etiology was made only on post mortem biopsy. We suggest that the presence of vascular thrombosis on CECT in a patient presenting with febrile illness should be regarded as an indicator of possible infection by angiotropic fungi, such as zygomycosis, which could help clinician to pursue histological diagnosis aggressively.

Case Report: Gastrointestinal Basidiobolomycosis in a Young Girl with Cystic Fibrosis and Celiac Disease.

Basidiobolomycosis is an uncommon fungal infection that has been reported in the literature mainly as a cause of infection in the skin and subcutaneous tissue. Intraabdominal infections have been reported in tropical and subtropical areas in the Middle East, such as Iran and Saudi Arabia, and in the United States. Our patient was a 6-year-old girl with cystic fibrosis and celiac disease who was referred to our department with a history of chronic abdominal pain. In the imaging studies of the abdomen and pelvis, a large retroperitoneal mass was reported in the right upper part of the abdomen with involvement of the duodenum and the mesentery of the small and large intestines, as well as the superior mesenteric vessels, and was diagnosed as basidiobolomycosis through biopsy. Because of the large unresectable mass, the patient was first treated with antifungal drugs for 2 months and then surgical resection was performed. The main point in the management of these patients is a combination of antifungal therapy and surgical resection. In some patients, complex surgeries such as the Whipple procedure may be performed to appropriately manage intraabdominal infections.

Eosinophilic granulomatous gastrointestinal and hepatic abscesses attributable to basidiobolomycosis and fasciolias: a simultaneous emergence in Iraqi Kurdistan.

BACKGROUND: Deep eosinophilic granulomatous abscesses, as distinguished from eosinophilic subcutaneous abscesses, are rare. Most reports are from the Far-East and India where the most commonly attributed cause is Toxocara. Sulaimaniyah in Northeastern Iraq has experienced an outbreak of eosinophilic granulomatous liver and gastrointestinal (GI) abscesses beginning in 2009. The purpose of this study was to determine the etiology and guide treatment. METHODS: The study was an ongoing investigation of patients having a histopathologic diagnosis of eosinophilic granulomatous abdominal abscesses in Sulaimaniyah hospitals from May 2009 to August 2012. Tissues were examined for organisms, and Enzyme Linked Immunoabsorbent Assays (ELISA) were performed for serum antibodies to Fasciola hepatica, Toxocara, and Echinococcus granulosus. RESULTS: Fourteen patients had granulomatous inflammation surrounding a central necrotizing eosinophilic exudate identified in surgical pathology specimens from abdominal surgeries. Two children and four adults had abscesses that formed GI masses. These patients included a 39 year old male with oropharyngeal and transverse colon disease, and a 48 year old male with liver and GI abscesses. All sites demonstrated a Zygomycete fungus surrounded by eosinophilic Splendori-Hoeppli material consistent with basidiobolomycosis. Five of the six patients with fungal disease were treated by surgery and 4 to 7 months of itraconozol. One child died of intestinal perforation while receiving IV amphotericin B; two adults required additional surgery for recurrent GI obstruction. Eight patients had isolated liver abscesses with no organisms identified by histopathology: ELISA results for F. hepatica were positive for five, borderline for one, and negative for two patients. These eight patients were treated for fascioliasis by surgical resection of localized abscesses and albendazol. One patient serologically positive for F. Hepatica was found to have a common duct fluke two years after initial diagnosis. Serological testing for Toxocara and Echinococcus granulosus was negative in all 14 patients. CONCLUSIONS: Basidiobolomycosis and F. hepatica are implicated as the cause of abdominal eosinophilic granulomatous abscesses in 12 of 14 patients identified over a period of 40 months in northern Iraq. Treatment was complicated by chronic biliary tract disease in fascioliasis and perforation and recurrent intestinal obstruction with basidiobolomycosis.

Fatal-mixed cutaneous zygomycosis-aspergillosis: a case report.

Mucorales and Aspergillus are molds responsible for infections in immunocompromised patients. In this report, we describe a case of a rare extensively mixed cutaneous infection caused by Lichtheimia ramosa, Aspergillus fumigatus and Aspergillus terreus in a neutropenic patient suffering from an acute leukemia. The fatal outcome of this patient can be attributed to its hematologic malignancy, the extensive nature of the lesions and the resistance of the strains to antifungals.

Into the winter whiteness.

A father recounts a transformative journey of love, loss, and learning as his son developed and eventually died of a fungal infection that occurred during a period of immunocompromise after a bone marrow transplantation to treat hypodiploid leukemia. He then thanks the many physicians, researchers, family members, and friends who came together to memorialize his son and create knowledge, hope, and wisdom, so that others will not have to suffer a similar journey.

Pulmonary zygomycosis in a non-neutropenic patient with myelodysplastic syndrome on lenalidomide.

Pulmonary zygomycosis is an uncommon infection that occurs mostly in immunocompromised patients. We report the case of a 75-year-old man with myelodysplastic syndrome, treated with lenalidomide for 3 months, who developed respiratory failure and a rapidly progressive left upper lobe consolidation. An extensive workup was unrevealing of the etiology, and the patient expired. A full autopsy was declined, but an in situ post-mortem transbronchial lung biopsy revealed pulmonary zygomycosis. This unique case illustrates the potential risks of lenalidomide therapy in patients with myelodysplastic syndrome and the difficulties in diagnosing pulmonary zygomycosis. To our knowledge this is the first report of a diagnostic in situ post-mortem transbronchial lung biopsy.

Isolated renal zygomycosis: novel diagnostic and prognostic criteria with experience of a tertiary care center.

BACKGROUND: Isolated renal zygomycosis is a life-threatening infection and difficult to diagnose ante mortem due to varied presentations. Most reports in the literature are case reports. We are presenting our experience of 10 patients. MATERIALS AND METHODS: Retrospective data of 10 consecutive patients with primary renal zygomycosis, including 2 post-transplant patients, in our tertiary care center was analyzed. Epidemiological characteristics, predisposing conditions, clinical presentation, diagnostic findings and treatment outcomes were recorded. Characteristic radiological findings were recorded. Localized disease was managed by supportive treatment or percutaneous drainage and extensive disease with unilateral or bilateral nephrectomy. Renal involvement was confirmed in all patients by histopathology. RESULTS: The mean age of presentation was 35 years. Five patients who had bilateral renal involvement presented with oliguric acute renal failure, hematuria and abdominal pain. Three had unilateral renal disease and presented with flank pain and fever. The two post-transplant patients presented with fever and graft dysfunction. Even after aggressive treatment 5 patients died, accounting for a mortality rate of 50%. CONCLUSION: Isolated renal zygomycosis can be diagnosed with typical radiological findings, combined with clinical, laboratory and histopathological features. This study describes the newer ante mortem radiological diagnostic criteria and prognostic predictors of the disease.

Invasive intracerebral fungal infection in a leukaemic patient.

Intracerebral abscesses secondary to Zygomycosis are potentially fatal. A 12-year-old girl with acute promyelocytic leukaemia was successfully treated for intracerebral abscess caused by Zygomycosis with complete surgical excision and combination antifungal therapy.

Disseminated nocardiosis masking an atypical zygomycosis presentation in a kidney transplant recipient.

Immunosuppressive agents increase the vulnerability of solid organ transplant patients to opportunistic infections. An atypical clinical presentation of a bacterial and fungal co-infection makes diagnosis and treatment even more challenging in this population. A 54-year-old hypertensive woman underwent a cadaveric kidney transplant after years on hemodialysis. Her treatment included mycophenolate, tacrolimus, and prednisone. By post-transplant week 8, she had pneumonia followed by progressive visual changes and seizures. Diagnostic work-up, consisting of magnetic resonance imaging of the brain and chest x-ray, showed several cerebral ring-enhancing lesions, and a pulmonary cavitary lesion. Disseminated nocardiosis was suspected and therapy was started. Skin biopsy was taken from a nodular lesion and culture confirmed Nocardia species infection. During hospitalization, neurological deficit persisted with worsening of brain lesions. She underwent excision of a brain abscess and the final pathologic report showed mucormycosis, revealing the patient's co-infection by 2 different pathogens. After therapy with liposomal amphotericin B and posaconazole, she has remained stable for more than 1 year. Disseminated nocardiosis masked and delayed the diagnosis and treatment of a more aggressive and worrisome organism. Mucormycosis, as a non-fatal isolated brain abscess without rhinal involvement, is an atypical presentation, and only a few cases have been reported.

Pneumonia and lung infections due to emerging and unusual fungal pathogens.

Invasive mold infections affecting the lungs are increasing in incidence and diversity. Severely immunocompromised patients are particularly vulnerable to infection from unusual, normally nonpathogenic fungi that are found naturally in the environment. Certain fungi such as Scedosporium and the dematiaceous fungi also cause lung disease in hosts without overt immune compromise. The impacts of these emerging pathogens range from airway colonization to locally invasive lung, and disseminated, disease. Diagnosis requires isolation and identification of the etiologic agent by either or both phenotypic and molecular biology methods. Evidence of tissue invasion on histopathology is often required to distinguish infection from colonization. Diagnostic imaging techniques are nonspecific, and there are no reliable serological biomarkers of infection. Many rare molds and yeasts demonstrate reduced in vitro susceptibility to antifungal agents. Although amphotericin B formulations remain clinically useful for many of these infections, voriconazole and posaconazole are more effective for some of these difficult-to-treat pathogens. Surgical resection of diseased tissue and support of the host immune system are often required to optimize outcomes.

Sudden vision loss caused by rhino-orbital zygomycosis in diabetic patients: case series.

Rhino-orbital zygomycosis is a life-threatening fungal infection generally occurring in patients with an underlying disorder, such as diabetes mellitus with ketoacidosis or with immunocompromising factors, although it may rarely appear in healthy individuals. The study has been undertaken to discuss the clinical presentation, pathogenesis, diagnostic work up and management of this rapidly progressive disease. Four male patients having uncontrolled diabetes and presenting with signs and symptoms of rhino-orbital zygomycosis were studied to illustrate the serious nature of the disease. All the four patients had rapidly deteriorating vision loss either unilateral or bilateral along with other nasal and orbital signs and symptoms. All the patients were put on liposomal amphotericin B and underwent orbital exenteration and pansinusectomy. One patient died, while the other three were successfully treated. Early diagnosis is critical in the prevention of morbidity and mortality associated with the disease. There is need for a high index of clinical suspicion in immunocompromised patients. Timely medical-surgical treatment proves extremely important for prognosis.

Rhinoentomophthoromycosis due to Conidiobolus coronatus. A case report and an overview of the disease in India.

Rhinoentomophthoromycosis due to Conidiobolus coronatus is a rare, chronic, granulomatous disease, occurring mainly in tropical Africa, South and Central America and south-east Asia, including India. We report a case of rhinoentomophthoromycosis in a 30-year-old male farmer, a resident of Gorakhpur city in Uttar Pradesh, which was diagnosed by histopathology and isolation C. coronatus in culture. The patient presented with a swollen nose with obstruction that had progressed slowly over one year. His nasal swelling was bilateral, diffuse, mildly tender, erythematous, non-pitting, with mucosal crusting and hypertrophy of inferior turbinates but no regional lympha-denopathy. A contrast-enhanced computed tomography (CECT) scan revealed bilateral pan-sinusitis with nasoethmoid polyposis. Culture of tissue from the nasal biopsy on Sabouraud glucose agar yielded multiple colonies of a mold with satellite smaller colonies at periphery. The isolate demonstrated the macroscopic and microscopic morphologic characteristics of C. coronatus. Its identity was further confirmed by direct DNA sequencing of internal transcribed spacer (ITS) and D1/D2 regions of rDNA. Haemotoxylin and eosin stained tissue sections of the skin biopsy revealed irregular epidermal acanthosis, marked inflammatory and granulomatous reaction with sparse, non-septate hyphae. The patient was treated successfully with a combination therapy of oral saturated potassium iodide solution, itraconazole, and intravenous infusion of amphotericin B. An overview of rhinoentomophthoromycosis cases reported to-date in India is presented.

[Case of a diabetic man cured of rhinocerebral zygomycosis]. / Rhinocerebralis zygomycosisból gyógyult diabeteses férfi esete.

UNLABELLED: Zygomycosis (mucormycosis) is a rare, highly aggressive opportunistic fungal disease caused by saprophytic fungi, belonging to the division Zygomycota, class Zygomycetes. Patients with immunodeficiency, neutropenia, iron overload, hematological malignancies, as well as diabetics with ketoacidosis are typically affected. CASE PRESENTATION: Authors describe the case of an eighteen-year old man with poor compliance suffering from diabetes since the age of nine. He was admitted with ketoacidotic somnolence in severe general condition with unilateral periorbital erythematous edema. Though from nasal exudates gained by the fibero-endoscopic sinus surgery Methicillin-resistant Staphylococcus aureus and Pseudomas aeruginosa was cultured, amphotericin-B was administered as the medical history, clinical picture suggested presence of zygomycosis. An invasive infection caused by Rhizopus oryzae was confirmed by histology and microbiology. DISCUSSION: The combination of antifungal therapy, repeated surgical interventions and granulocyte colony-stimulating factor resulted in good clinical response. Four month after discharge he is alive and doing well.

Entomophthoromycosis in a child: Delayed diagnosis and extensive involvement.

Entomophthoromycosis is a rare fungal infection of the skin and subcutaneous tissue occurring predominantly in tropical and subtropical regions. In children, it mostly affects the lower half of the body. With this, we report a case of Entomophthoromycosis in a 6-year-old girl who presented late with extensive involvement of the upper half of the body. She responded well to treatment with potassium iodide and itraconazole. We also reviewed cases of Entomophthoromycosis reported in children.