RESUMO
BACKGROUND AND OBJECTIVES: Zygomycosis, a severe form of fungal infection, is classified into two categories: Mucorales and Entomophthorales. Within the Entomophthorales category, Basidiobolomycosis is a rarely recognized genus that can have significant health implications. Prompt diagnosis and appropriate treatment, which includes the use of antifungal medication and surgical procedures, are vital for enhancing the prognosis of patients. The objective of this study is to investigate the response to treatment in patients hospitalized due to basidiobolomycosis. METHODS: We carried out a retrospective study, in which we analyzed data from 49 patients who were diagnosed with Entomophthorale, Zygomycosis, and Basidiobolomycosis at Namazi Hospital, Shiraz, between the years 1997 and 2019. The data included parameters such as demographic information, clinical symptoms, imaging findings, treatment methods, and patient outcomes. RESULTS: Out of 49 patients, 24 children, predominantly male (83.3%), were definitively diagnosed with basidiobolomycosis. The ages of the patients ranged from 1 to 16 years, with an average of 5.75 years. The most frequently observed clinical manifestations included abdominal pain (70.8%), fever (54.2%), hematochezia (41.7%), vomiting (20.8%), and anorexia (16.7%). Half of the patients exhibited failure to thrive (FTT), while abdominal distension was present in 25% of the cases, and a palpable abdominal mass was found in 37% of the patients. The primary treatment strategy incorporated surgical interventions complemented by a comprehensive antifungal regimen. This regimen included medications such as amphotericin B, cotrimoxazole, itraconazole, potassium iodide, and voriconazole. These were mainly administered in a combination therapy pattern or as a monotherapy of amphotericin B. Twenty-two patients were discharged, while two patients died due to complications from the disease. CONCLUSION: Our findings indicate that the prevailing treatment modalities generally involve surgical intervention supplemented by antifungal regimens, including Amphotericin B, Cotrimoxazole, Potassium Iodide, and Itraconazole.
Assuntos
Antifúngicos , Entomophthorales , Zigomicose , Humanos , Zigomicose/tratamento farmacológico , Zigomicose/microbiologia , Masculino , Antifúngicos/uso terapêutico , Feminino , Estudos Retrospectivos , Adolescente , Criança , Pré-Escolar , Lactente , Resultado do TratamentoRESUMO
Basidiobolomycosis is an uncommon fungal infection caused by the genus Basidiobolus. In immunocompetent children, it usually causes cutaneous infection and rarely affects the gastrointestinal tract, and it is extremely rare for the disease to spread. The present study reports the first case of disseminated basidiobolomycosis caused by Basidiobolus omanensis in a child with acute lymphoblastic leukemia who died as a result of uncontrolled infection and multi-organ failure despite surgical and antifungal therapy with L-AMB and voriconazole. A review of the literature yielded 76 cases, including the current case with the majority of which were reported as invasive gastrointestinal infection. The median age was 4 years (61 male and 15 female) and the majority of these children were from the Middle East (80%), specifically Saudi Arabia (45%). Most patients were treated with systemic antifungal agents (mostly itraconazole and amphotericin B). Surgical intervention was done in 25% of these patients and the death rate was 12%.
Assuntos
Entomophthorales , Leucemia-Linfoma Linfoblástico de Células Precursoras , Zigomicose , Criança , Humanos , Feminino , Masculino , Pré-Escolar , Zigomicose/diagnóstico , Zigomicose/tratamento farmacológico , Leucemia-Linfoma Linfoblástico de Células Precursoras/complicações , Itraconazol/uso terapêuticoRESUMO
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.
Assuntos
Doença Celíaca , Fibrose Cística , Infecções Intra-Abdominais , Zigomicose , Humanos , Feminino , Criança , Antifúngicos/uso terapêutico , Doença Celíaca/complicações , Doença Celíaca/diagnóstico , Fibrose Cística/complicações , Fibrose Cística/tratamento farmacológico , Zigomicose/complicações , Zigomicose/diagnóstico , Zigomicose/tratamento farmacológico , Infecções Intra-Abdominais/tratamento farmacológicoRESUMO
La conidiobolomicosis es una micosis subcutánea causada por un hongo saprofito, Conidiobulus spp. perteneciente a la clase Zigomicetos, orden Entomoftorales, que habita en regiones tropicales. La manifestación clínica clásica es la deformidad progresiva de estructuras faciales y su diagnóstico se basa en cultivos de la zona afectada y el estudio histopatológico, siendo el "fenómeno de Splendore-Hoeppli" el hallazgo más característico. Dada su baja frecuencia de presentación, no existe consenso sobre la mejor opción y tiempo de tratamiento. Aquí presentamos un caso de entomoftoromicosis rinofacial causada por Conidiobolus coronatus en un paciente inmunocompetente de la región sur de Colombia.
Conidiobolomycosis is a subcutaneous mycosis caused by a saprophytic fungus, Conidiobulus, belonging to the class of Zygomycetes, an order of Entomophtorales that inhabits tropical regions. Its most frequent clinical manifestation is the progressive deformity of facial midline structures, and the diagnosis is based on cultures taken from the affected area and the histopathological study, being the "Splendore-Hoeppli phenomenon" the most characteristic finding. Due to its low frequency of presentation, there is no consensus about the best option and treatment time. We present a case of rhinofacial entomophthoromycosis caused by Conidiobolus coronatus in an immunocompetent patient from the southern region of Colombia.
Assuntos
Humanos , Masculino , Adulto Jovem , Zigomicose/microbiologia , Zigomicose/diagnóstico por imagem , Imageamento por Ressonância Magnética , Conidiobolus/isolamento & purificação , Zigomicose/patologia , Zigomicose/tratamento farmacológico , Antifúngicos/uso terapêuticoRESUMO
Gastrointestinal basisdiobolomycosis is a rare fungal infection prevalent in the tropical and subtropical regions. It has variable clinical manifestations that present a challenge for timely diagnosis. Herein, we present a case of gastrointestinal basidiobolomycosis in a 34-year-old male. To the best of our knowledge, it is the first reported case of gastrointestinal basidiobolomycosis from Pakistan. The patient presented with abdominal pain and was first operated for perforated appendix and then for a mesenteric mass based on the findings of CT scan. Histopathologic examination showed broad septate fungal hyphae surrounded by eosinophilic proteinaceous material (Splendore-Hoppeli phenomenon), neutrophils, and histiocytes. Diagnosis of gastrointestinal basidiobolomycosis was established on the basis of this morphology.
Assuntos
Apendicite , Zigomicose , Masculino , Humanos , Adulto , Zigomicose/diagnóstico , Zigomicose/tratamento farmacológico , Dor Abdominal/etiologia , ConhecimentoRESUMO
Zygomycosis is a fungal infection in humans caused by orders Mucorales and Entomophthorales. The incidence of Mucorales causing mucormycosis is on a rise and is well documented, whereas Entomophthorales is rare. Among Entomophthorales, infections caused by Conidiobolus are more common than Basidiobolus Here we present a case of subcutaneous basidiobolomycosis in a female patient. The patient had hyperpigmentation in the thigh region for 6 months and serous discharge for 4 months. All initial findings suggested the inflammatory stage of morphea. Differential diagnoses of granuloma annulare, malignant melanoma and morphea were considered radiologically. A good suspicion of fungal aetiology by the dermatologist led to an appropriate diagnosis of subcutaneous basidiobolomycosis based on fungal culture and histopathological examination. Based on macroscopic and microscopic findings, the causative organism was confirmed to be Basidiobolus ranarum The patient was started on oral potassium iodide and itraconazole and showed a good prognosis.
Assuntos
Entomophthorales , Mucormicose , Esclerodermia Localizada , Dermatopatias Infecciosas , Zigomicose , Humanos , Feminino , Zigomicose/diagnóstico , Zigomicose/tratamento farmacológico , Zigomicose/microbiologia , Mucormicose/diagnóstico , Mucormicose/tratamento farmacológicoRESUMO
Entomophthoramycosis can be found in subtropical and tropical regions. This case illustrates common clinical features of conidiobolomycosis. Although this disease is not common, physicians working in these regions should be familiar with the clinical manifestations to enable early diagnosis and treatment.
Assuntos
Conidiobolus , Zigomicose , Humanos , Tela Subcutânea , Zigomicose/diagnóstico , Zigomicose/tratamento farmacológicoRESUMO
This is a case report of Basidiobolomycosis in a 65-year-old male patient from Jizan presenting with colonic perforation and concomitant liver involvement from February 2021 to July 2021. To control the infection, the patient underwent colonic resection and segmental liver resection, as well as three antifungal drugs. The treatment was successful, and the condition was completely resolved.
Assuntos
Entomophthorales , Perfuração Intestinal , Zigomicose , Idoso , Antifúngicos/uso terapêutico , Humanos , Perfuração Intestinal/cirurgia , Masculino , Zigomicose/diagnóstico , Zigomicose/tratamento farmacológico , Zigomicose/cirurgiaRESUMO
Conidiobolomycosis caused by Conidiobolus species is an uncommon infection restricted to tropical and subtropical regions, usually affecting immunocompetent individuals. More than half of pediatric cases of conidiobolomycosis across the globe are from India. We report a case of subcutaneous conidiobolomycosis in an adolescent with development delay who responded to combined therapy with itraconazole and saturated solution of potassium iodide.
Assuntos
Conidiobolus , Zigomicose , Adolescente , Antifúngicos/uso terapêutico , Criança , Humanos , Itraconazol/uso terapêutico , Zigomicose/diagnóstico , Zigomicose/tratamento farmacológicoRESUMO
BACKGROUND: Conidiobolomycosis is a rare tropical rhinofacial fungal infection which has not been well characterised. The available evidence in its management is sparse due to lack of clinical studies and the limited data on antifungal susceptibility patterns. OBJECTIVE: To analyse the clinical manifestations, antifungal treatment and outcomes of patients with conidiobolomycosis and to determine antifungal susceptibility profiles of the isolates. PATIENTS/METHODS: Retrospective analysis of data of all patients with a diagnosis of conidiobolomycosis confirmed by histopathology and culture at a tertiary care hospital from 2012 to 2019 was done. RESULTS: There were 22 patients, 21 males and one female, with a mean age of 37.1 years. Most common presenting symptom was nasal obstruction, found in 20 (90.90%) patients. Patients who presented within 12 months had a better cure rate (85%) compared to those who presented late (67%). Among the 19 patients who had a follow-up, good outcome was seen in 15 of the 17 (88.24%) patients who were on itraconazole or potassium iodide containing regimen. Of the six patients who received additional trimethoprim-sulphamethoxazole (co-trimoxazole), 67% showed good outcome with two patients showing complete cure and two patients still on treatment with significant improvement. High minimum inhibitory concentration (MIC) values were noted for azoles and amphotericin B, whereas co-trimoxazole showed lowest MIC ranges. CONCLUSION: Itraconazole and potassium iodide are reasonable first-line options for the treatment of conidiobolomycosis. Good clinical response to KI and comparatively lower MIC of co-trimoxazole are promising. Further studies are required for developing clinical breakpoints that can predict therapeutic outcomes.
Assuntos
Antifúngicos/uso terapêutico , Conidiobolus/efeitos dos fármacos , Doenças Raras/microbiologia , Zigomicose/tratamento farmacológico , Zigomicose/microbiologia , Adulto , Gerenciamento Clínico , Face/microbiologia , Face/patologia , Feminino , Humanos , Masculino , Testes de Sensibilidade Microbiana , Pessoa de Meia-Idade , Doenças Nasais/tratamento farmacológico , Doenças Nasais/microbiologia , Doenças Raras/tratamento farmacológico , Estudos Retrospectivos , Centros de Atenção Terciária/estatística & dados numéricos , Adulto JovemRESUMO
Conidiobolomycosis is a neglected tropical fungal infection involving the head and neck region. Here we report the first case of atypical conidiobolomycosis presenting with dysphagia and significant weight loss from Odisha, India. It was diagnosed by histopathology and fungal culture and was suscessfully treated with saturated solution of potassium iodide.
Assuntos
Transtornos de Deglutição , Zigomicose , Antifúngicos/uso terapêutico , Conidiobolus , Transtornos de Deglutição/tratamento farmacológico , Transtornos de Deglutição/etiologia , Humanos , Índia , Zigomicose/diagnóstico , Zigomicose/tratamento farmacológicoRESUMO
Entomophthoramycosis is a rare fungal infection of nose, paranasal sinuses and subcutaneous tissues found in tropical and subtropical region. From India very few cases have been reported. Here we report a case of Entomophthoramycosis due to Conidiobolus coronatus from the eastern India who presented with slowly growing rhinofacial swelling and right sided nasal obstruction due to intranasal mass. The case was diagnosed by typical histopathological findings of broad aseptate hyphae with surrounding eosinophilic granular material (Splendore Hoeppli phenomenon) on microscopy of nasal biopsy material and confirmed by PCR assay of DNA and sequencing from biopsy tissue. Treatment with saturated solution of potassium iodide and itraconazole was successful and clinical cure was attained in 8 months.
Assuntos
Antifúngicos , Zigomicose , Antifúngicos/uso terapêutico , Biópsia , Face , Humanos , Índia , Zigomicose/diagnóstico , Zigomicose/tratamento farmacológicoRESUMO
Conidiobolomycosis is an uncommon, chronic, localized subcutaneous mycosis primarily affecting rhinofacial region. It is reported mainly from tropical and subtropical countries. The condition is underreported due to the lack of clinical suspicion and usually mismanaged. This rare mycosis is due to the genus Conidiobolus within the order Entomophthorales of class Zygomycetes. Here we present 3 cases of rhinofacial conidiobolomycosis in otherwise healthy adults from different parts of Sri Lanka over 1-year period. All patients had disfiguring subcutaneous lesions in the rhinofacial area. The diagnoses were based on isolation of Conidiobolus coronatus in clinical specimens.
Assuntos
Conidiobolus/isolamento & purificação , Dermatomicoses/diagnóstico , Dermatoses Faciais/diagnóstico , Zigomicose/diagnóstico , Adolescente , Adulto , Idoso , Antifúngicos/uso terapêutico , Dermatomicoses/tratamento farmacológico , Dermatomicoses/microbiologia , Dermatomicoses/patologia , Dermatoses Faciais/tratamento farmacológico , Dermatoses Faciais/microbiologia , Dermatoses Faciais/patologia , Humanos , Masculino , Nariz/microbiologia , Nariz/patologia , Zigomicose/tratamento farmacológico , Zigomicose/microbiologia , Zigomicose/patologiaRESUMO
Basidiobolomycosis is a fungal infection caused mainly by Basidiobolus ranarum, a filamentous fungus of the order Entomophthorales and the family Basidiobolaceae. This infection typically involves the skin and soft tissue; however, visceral organ involvement has also been reported. Here, we report a case of gastrointestinal basidiobolomycosis in a young child who presented with acute bloody diarrhea which was initially misdiagnosed as intussusception.
Assuntos
Entomophthorales , Gastroenteropatias , Zigomicose , Antifúngicos/uso terapêutico , Criança , Diarreia/tratamento farmacológico , Diarreia/etiologia , Gastroenteropatias/tratamento farmacológico , Humanos , Lactente , Doenças Raras/tratamento farmacológico , Zigomicose/diagnóstico , Zigomicose/tratamento farmacológicoRESUMO
INTRODUCTION: For the best management of the zygomycosis in immunocompromised patients, the present study aims to detect and identify the etiologic agents by DNA sequencing method and their related factors in clinical samples of patients. METHODOLOGY: Clinical samples from 1,058 patients admitted in 11 university hospitals in Shiraz, Southern Iran were collected between July 2015 and July 2018. All samples (bronchoalveolar lavage, sputum, blood, tissue) were examined by routine microscopic and culture tests for zygomycetes. The etiologic agents were identified by the molecular method and sequencing. RESULTS: Direct microscopic examinations or pathology smear, culture, and PCR were positive in 61 (5.8%), 15 (1.4%), and 103 (9.7%) patients, respectively. According to EORTC/MSG criteria, the rates of proven, probable, and possible zygomycosis were 59.2% (61/103), 14.6% (15/103), and 26.2% (27/103 patients), respectively. The most prevalent etiologic agents according to sequencing were Rhizopus oryzae (44 cases), Rhizopus microsporus (31 cases), Rhizopus stolonifer (15 cases). Twenty-two patients (21.4%) with positive PCR died. There were significant relations between zygomycosis and the underlying disease (p = 0.043) and prior antifungal therapy (p = 0.023). White blood cell count was in the normal range in 14.1% of patients, and the means of erythrocyte sedimentation rate (ESR) and C reactive protein (CRP) were 65 mm/hour and 57 mg/L, respectively. CONCLUSIONS: Molecular methods and sequencing may have considered as suitable tools to diagnose zygomycosis. Identification of the etiologic agents may be considered as the future antifungal therapy and management of the respective patients.