Intractable Nocardial mycetoma with possible colonisation by Candida species.

A Japanese male in his 30s with no underlying medical condition presented with painless nodules after being bitten by a dog during a stay in Bali, Indonesia, 7 years earlier. He was referred to our department with multiple ulcers, nodules, and masses on the right leg. The final diagnosis was mycetoma caused by Nocardia vulneris, which may have been exacerbated by colonization of Candida parapsilosis and C. tropicalis as these yeasts were isolated by culture from the tissue. Treatment with minocycline hydrochloride and sulfamethoxazole trimethoprim showed partial efficacy, but the addition of posaconazole achieved significant efficacy. This suggests that the surmised coexistence of pathogenic yeasts of lower virulency may have made mycetoma in this case intractable.

Towards enhanced control of mycetoma: a roadmap to achieve the UN's sustainable development goals by 2030.

Mycetoma is a neglected tropical disease (NTD) with devastating morbidity and stigma. Despite increased awareness and international collaboration, the burden of mycetoma is largely unknown and diagnosis and treatment are difficult. Addressing mycetoma globally aligns with several United Nation's Sustainable Development Goals (SDGs). Little progress has been made since the WHO's NTD roadmap publication in 2020. The Global Mycetoma Working Group proposes an enhanced mycetoma-control roadmap to meet the SDGs, stimulate progress and improve the lives of patients experiencing mycetoma. By aligning mycetoma management with the goals and targets of this enhanced roadmap, it becomes possible to leverage existing resources, infrastructure and partnerships to improve the lives of affected individuals and communities. This updated assessment is designed for the benefit of health workers and providers in mycetoma-endemic areas, NTD government officials, civil society and funding and implementing agencies.

Dot in circle sign on MRI in foot mycetoma.

Mycetoma or Maduramycosis is a chronic granulomatous infectious condition encountered mostly in tropical and subtropical regions. It affects the deep subcutaneous tissues, which may progress to involve the muscles and bones later in the course of the disease. It can be caused by fungi (eumycetoma), and bacteria (actinomycetoma) predominantly affecting the foot. Demonstration of the causative agent by biopsy and microbiological studies helps to establish a confirmative diagnosis, and choosing correct antimicrobial therapy. However, it may be delayed resulting in increased patient morbidity. Thus, imaging plays a vital role in early recognition & prompt treatment, especially MRI which is a non-invasive procedure demonstrating the hallmark dot in circle sign. Here we report a case of mycetoma foot with pathognomic MRI findings.

Dermoscopy aids in lesion selection for microbiological and histopathological assessment of mycetoma.

Mycetoma, a chronic granulomatous infection of the skin and the subcutaneous tissue, is characterized by discharge of exudate containing grains. This report illustrates the importance of dermoscopy in selecting lesions for both microbiological and histopathological assessment, which are considered the gold standard of diagnosis in mycetoma.

Outcomes of pulmonary resection in pulmonary aspergilloma: A retrospective cohort study in a single tertiary-care hospital in Northern Thailand.

BACKGROUND: To date, surgery is the best approach to maximize a cure for symptomatic aspergilloma, but this is not without risk of both morbidity and mortality. The aim of this study is to present the characteristics and outcomes of 77 patients diagnosed with aspergilloma who underwent surgery at Chiang Mai University Hospital (CMUH), and to identify risk factors for composite major postoperative complications (CMPC). METHODS: This is an observational retrospective cohort study carried out at CMUH over a period of 11 years from January 1, 2010, to February 28, 2021. Patient characteristics and postoperative outcomes were studied. The primary outcomes were categorized into CMPC. Univariable and multivariable risk regression analysis were used to identify risk factors of CMPC, with risk ratio (RR) and 95% confidence intervals being calculated. RESULTS: There were 77 patients included in this study; 27 patients identified as having CMPC and 55 patients as a non-CMPC group. From the multivariable analysis, a factor associated with CMPC included perioperative FFP transfusion (risk ratio (RR) 1.01,95 % CI 1.01-1.02) and preoperative angiogram embolization (RR 8.42, 95 % CI 1.44-49.06) whereas immediate extubation (RR 0.22, 95% CI 0.06-0.81) was less likely to be associated with CMPC. There was a trend of increased risk of CMPC in patients received perioperative blood transfusion, but the data did not reach statistical significance. CONCLUSIONS: This study has identified a need for patient profiling before embarking on lung surgery for aspergilloma, to predict outcomes and allocate resources appropriately for safer surgery.

Mycetoma in Ecuador.

BACKGROUND: Previous studies on the distribution of mycetoma globally have failed to identify Ecuador as an endemic country. METHODS: We present data on 35 cases of mycetoma in Ecuador between 1955 and 2021: 5 cases from our experience and 30 cases from the literature. RESULTS: Eight cases of eumycetoma (23%) and 27 cases of actinomycetoma (77%) were diagnosed. Most cases originated from the coastal region of Ecuador. CONCLUSIONS: For the first time in an English-language publication, this communication confirms the presence of mycetoma in Ecuador, securing Ecuador's position on the global mycetoma map.

Cutaneous Hyalohyphomycosis and Its Atypical Clinical Presentations in Immunosuppressed Patients.

There has been a substantial increase in the number of cases of invasive fungal infections worldwide, which is associated with a growing number of immunosuppressed patients and a rise in antifungal resistance. Some fungi that were previously considered harmless to humans have become emerging pathogens. One of them is Purpureocillium lilacinum, a ubiquitous filamentous fungus commonly found in the environment, especially in the air and soil. P. lilacinum belongs to a bigger group of hyaline fungi that cause hyalohyphomycosis, a fungal infection caused by fungi with colorless hyphae. Although this is a heterogeneous group of fungi, there are similarities regarding their ubiquity, ways of transmission, affected patients, and difficulties in diagnostics and treatment. In hyalohyphomycosis, the skin is one of the most affected organs, which is why the involvement of dermatologists is crucial for the initial assessment, since the timely recognition and early diagnosis of this condition can prevent life-threatening infections and death. In this review, we covered cutaneous hyalohyphomycosis caused by P. lilacinum and other fungi in the same group, including Fusarium, Penicilium, Scedosporium, Scopulariopsis, Acremonium, and Trichoderma genera.

Actinomycetoma by Cellulosimicrobium cellulans in a Young Man from Guinea-Bissau: Short Literature Review Regarding a Case Report.

Mycetoma is caused by the subcutaneous inoculation of filamentous fungi or aerobic filamentous bacteria. Cellulosimicrobium cellulans is a gram-positive bacterium from the order Actinomycetales that rarely causes human disease. The diagnosis is based on the clinical presentation and identification of the causative microorganism. We present a short literature review regarding the case report of a young man diagnosed with actinomycetoma due to Cellulosimicrobium cellulans and who received treatment with an association of amikacin and sulfamethoxazole/ trimethoprim (Welsh regimen).

Mycetoma case series in Somalia.

Mycetoma is a suppurative chronic bacterial or fungal disease inoculated into the body by minor trauma which may penetrate from subcutaneous tissue to bone. Although the lower extremities are most commonly affected, rare forms can also be seen from time to time. The diagnostic triad of swelling in the affected area, multiple sinus formation, and purulent discharge with grains are typical. Definitive diagnosis is made by isolation of the causative pathogen, radiologic imaging, and histopathologic examination. Antifungal and antibacterial options are applied together with surgery. Our aim in this case series is to report and analyze 10 rare cases of mycetoma.

Invasive Eumycotic mycetoma of the nasal septum in a post-dialysis patient: A case report.

BACKGROUND: Mycetoma is a slowly progressive chronic granulomatous disease of the skin, subcutaneous tissue, and underlying or adjacent cartilage or bone. Most commonly involves the foot region. Other parts such as the knee, arm, leg, head, neck, thigh, perineum, chest, abdominal walls, facial bones, mandible, paranasal sinuses, eyelid, vulva, orbit, and scrotum are seldom affected. METHODS: This is a rare presentation of Eumycotic mycetoma involving the nasal septum. Surgical debridement is done under local anesthesia. Histopathological examination of debrided specimen guided in the diagnosis and treatment. RESULTS: Histopathological examination is the one that confirms the diagnosis and rules out the other granulomatous conditions and fungal rhinitis causing septal perforation. CONCLUSIONS: In an immunocompromised state, we know that mucormycosis and zygomycosis are known to cause aggressive complications like orbital invasion and palatal perforation by vascular route. However, other fungal infections also can lead to septal perforations whenever there is lessened resistance by the mucosal barrier due to trauma (nasal intubations).

Evaluation of a computational model for mycetoma-causative agents identification.

BACKGROUND: The therapeutic strategy for mycetoma relies heavily on the identification of the causative agents, which are either fungal or bacterial. While histopathological examination of surgical biopsies is currently the most used diagnostic tool, it requires well-trained pathologists, who are lacking in most rural areas where mycetoma is endemic. In this work we propose and evaluate a machine learning approach that semi-automatically analyses histopathological microscopic images of grains and provides a classification of the disease as eumycetoma or actinomycetoma. METHODS: The computational model is based on radiomics and partial least squares. It is assessed on a dataset that includes 890 individual grains collected from 168 patients originating from the Mycetoma Research Centre in Sudan. The dataset contained 94 eumycetoma cases and 74 actinomycetoma cases, with a distribution of the species among the two causative agents that is representative of the Sudanese distribution. RESULTS: The proposed model achieved identification of causative agents with an accuracy of 91.89%, which is comparable to the accuracy of experts from the domain. The method was found to be robust to a small error in the segmentation of the grain and to changes in the acquisition protocol. Among the radiomics features, the homogeneity of mycetoma grain textures was found to be the most discriminative feature for causative agent identification. CONCLUSION: The results presented in this study support that this computational approach could greatly benefit rural areas with limited access to specialized clinical centres and also provide a second opinion for expert pathologists to implement the appropriate therapeutic strategy.

Environmental detection of eumycetoma pathogens using multiplex real-time PCR for soil DNA in Sennar State, Sudan.

BACKGROUND: Mycetoma is a chronic disease affecting the skin and subcutaneous tissue endemic in the tropical and subtropical regions. Several bacteria and fungi can cause mycetoma, but fungal mycetoma (eumycetoma) is challenging because the treatment requires a combination of a long-term antifungal agent and surgery. Although the transmission route has not yet been elucidated, infection from the soil is a leading hypothesis. However, there are few soil investigation studies, and the geographical distribution of mycetoma pathogens is not well documented. Here, we used multiplex real-time PCR technology to identify three fungal species from soil samples. METHODS: In total, 64 DNA samples were extracted from soil collected in seven villages in an endemic area in Sennar State, Sudan, in 2019. Primers and fluorescent probes specifically targeting the ribosomal DNA of Madurella mycetomatis, Falciformispora senegalensis, and F. tompkinsii were designed. RESULTS: Multiplex real-time PCR was performed and identified the major pathogen, M. mycetomatis that existed in most sites (95%). In addition, two other pathogens were identified from some sites. This is the first report on the use of this technique for identifying the eumycetoma causative microorganisms. CONCLUSIONS: This study demonstrated that soil DNA investigation can elucidate the risk area of mycetoma-causative agents. The results will contribute to the design of prevention measures, and further large-scale studies may be effective in understanding the natural habitats of mycetoma pathogens.

The combination of manogepix and itraconazole is synergistic and inhibits the growth of Madurella mycetomatis in vitro but not in vivo.

Mycetoma is a neglected tropical disease commonly caused by the fungus Madurella mycetomatis. Standard treatment consists of extensive treatment with itraconazole in combination with surgical excision of the infected tissue, but has a low success rate. To improve treatment outcomes, novel treatment strategies are needed. Here, we determined the potential of manogepix, a novel antifungal agent that targets the GPI-anchor biosynthesis pathway by inhibition of the GWT1 enzyme. Manogepix was evaluated by determining the minimal inhibitory concentrations (MICs) according to the CLSI-based in vitro susceptibility assay for 22 M. mycetomatis strains and by in silico protein comparison of the target protein. The synergy between manogepix and itraconazole was determined using a checkerboard assay. The efficacy of clinically relevant dosages was assessed in an in vivo grain model in Galleria mellonella larvae. MICs for manogepix ranged from <0.008 to >8 mg/l and 16/22 M. mycetomatis strains had an MIC ≥4 mg/ml. Differences in MICs were not related to differences observed in the GWT1 protein sequence. For 70% of the tested isolates, synergism was found between manogepix and itraconazole in vitro. In vivo, enhanced survival was not observed upon admission of 8.6 mg/kg manogepix, nor in combination treatment with 5.7 mg/kg itraconazole. MICs of manogepix were high, but the in vitro antifungal activity of itraconazole was enhanced in combination therapy. However, no efficacy of manogepix was found in an in vivo grain model using clinically relevant dosages. Therefore, the therapeutic potential of manogepix in mycetoma caused by M. mycetomatis seems limited.

Treatment of Madurella mycetomatis-caused mycetoma consists of extensive exposure to antifungals and surgery. To improve therapy, we evaluated manogepix, a novel antifungal agent, as a therapeutic option against M. mycetomatis. Our findings suggest limited therapeutic potential for manogepix.

Actinomycosis of the sole of the foot (Madura foot): A rare case report and literature review.

INTRODUCTION AND IMPORTANCE: Actinomycosis of the foot is a rare, chronic, granulomatous infectious ailment affecting the skin, dermis, and subcutaneous tissues. It is caused by a fungal or actinomycotic agent, resulting in a pseudotumoral appearance. This pathology typically progresses slowly, leading to delayed diagnosis. The primary objective of this report is to emphasize the rarity and clinical significance of this highly uncommon medical condition. CASE PRESENTATION: We present the case of a 45-year-old woman with a mass on the sole of her right foot. The condition was treated through excisional biopsy along with combined antibiotic therapy. Histopathology of the excised mass confirmed the presence of actinomycosis, ultimately confirming the diagnosis. The patient's progress was satisfactory, with no recurrence observed over a 2-year follow-up period. CLINICAL DISCUSSION: Actinomycosis of the foot is a common pathology in tropical and subtropical regions where climatic conditions promote its emergence. It is a chronic, granulomatous infection of exogenous origin, caused by either fungal (eumycetoma) or bacterial (actinomycetoma) agents. When left unrecognized, these infections can lead to complications, including osteoarticular lesions, with a severe impact on the functional prognosis. The presence of foot swelling should raise suspicion of mycetoma as a diagnosis, with confirmation relying on histological analysis. Management typically involves the aggressive control of invasive soft-tissue masses, often followed by prolonged antibiotic treatment. Skin grafting is a standard method for closing Madura foot defects, and continuous surveillance is necessary due to the potential for actinomycosis recurrence. CONCLUSION: In the context of Morocco, actinomycosis of the foot remains sporadic. Identifying these conditions presents significant diagnostic challenges, leading to unfortunate treatment delays. These largely unrecognized conditions have the potential to become more complex by causing osteoarticular damage and lesions, ultimately worsening the functional outlook. Early vigilance is advisable upon observing foot swelling, considering the possibility of actinomycosis of the foot. A definitive diagnosis requires histological analysis, a critical step.

Unusual manifestation of cystic mycetoma lesions: A case report.

Key Clinical Message: This case presents an atypical cystic presentation of mycetoma without sinuses or discharge. Awareness of these variations is crucial for accurate diagnosis and timely intervention, highlighting the need for healthcare professionals to consider diverse manifestations of mycetoma. Abstract: Mycetoma is a chronic and debilitating infectious disease characterized by localized swellings and granulomatous lesions. It primarily affects individuals in tropical and subtropical regions and is caused by certain fungi or bacteria. While mycetoma typically presents with sinuses and discharge, this case report presents a unique cystic presentation without these features. The patient, a 12-year-old female from Sudan, presented with a painless swelling on the dorsum of her right foot. Physical examination revealed a round, non-tender, and fluctuant mass. Histopathological examination confirmed actinomycetoma caused by Streptomyces somaliensis. The patient was successfully treated with a combination of antibiotherapy. This atypical presentation underscores the need for healthcare professionals to consider uncommon variations of mycetoma for accurate diagnosis and management.

Case report: The dot in circle sign: A pathognomonic MRI sign of Madura foot.

Mycetoma or Madura's foot is a rare condition in Morocco. It is a chronic infection that affects deep subcutaneous tissues and may eventually spread to deep tissues, muscles, and bones. It is caused by fungi or bacteria and most often affects the foot. Biopsy and microbiological examination help to establish a definitive diagnosis, but may delay it. Imaging plays a crucial role in early diagnosis, particularly MRI findings that show a pathognomonic sign of mycetoma which is the dot-in-the-circle sign. Computed tomography provides a better analysis of bone changes than plain radiography. Treatment is very difficult, especially if the diagnosis is delayed, and may even lead to amputation. We present here a case of mycetoma of the foot with some MRI features.

A Rare Case of Oral Cavity Eumycetoma.

Rationale: Mycetoma is a chronic granulomatous infection that involves the skin and subcutaneous tissue which presents as an abscess with multiple draining sinuses. It is common in the tropical and subtropical regions where the conditions favour the growth of the soil saprophytes - fungi and bacteria. Patient Concerns: We report the rare case of a 62-year-old patient who presented with a soft-tissue overgrowth in the mandibular posterior ridge area for eight months. Diagnosis: The patient was diagnosed with eumycetoma, with foreign-body reaction based on clinical and histopathological examination which revealed characteristic brown-coloured amorphous grains. Treatment: The lesion was surgically excised under local anaesthesia. Outcomes: On the seventh day post-operative follow-up, the patient was relieved of pre-operative symptoms and the surgical site had healed well. Take-Away Lessons: This particular case of eumycetoma had an unusual site and appearance, making the clinical diagnosis confusing with other reactionary lesions of the oral cavity.

Improving mycetoma case detection through the training of community health workers in Northern Uganda: protocol for a stepped-wedge cluster-randomized trial.

Introduction: The World Health Organization acknowledges the need for countries to incorporate neglected tropical disease care into their routine health care system. However, low detection rates and late presentation of mycetoma to health facilities have been observed in endemic countries, including Uganda. Objective: To empower community health workers (CHWs) in Northern Uganda to recognize and refer suspects of mycetoma to health facilities. Design: This will be a stepped-wedge cluster-randomized trial based in Gulu and Pader districts over a period of 9 months with sequential crossover from intervention phase to the control phase at different time points until both districts are exposed to the intervention. Methods and Analysis: The study will leverage on the ongoing partnership between Northern Uganda Medical Mission and the Uganda Ministry of Health that has trained over 300 CHWs in Gulu and Pader. The study evaluation will be done using the RE-AIM (Reach, Effectiveness, Adoption, Implementation, and Maintenance) framework. The expected outcome of the study is increased detection and referral of suspects of mycetoma. Data will be analyzed using STATA 17.0 and Friedman statistics or Analysis of Variance to determine increase in case identifications and referrals. Ethics and Registration: The study was approved by Mulago Hospital Research and Ethics Committee (MHREC 2406) and registered with Pan African Clinical Trial Registry (PACTR202301534749787). Dissemination: The results from this trial will be published in a peer-reviewed journal. In addition, the findings will be shared at conferences, with funders, and at other research meetings.

Sinonasal Oxalosis due to Fungal Rhinosinusitis: A Unique Case of a Destructive Pseudotumor.

Oxalosis refers to the accumulation of calcium oxalate crystals in various organs and tissues, most commonly due to Aspergillus infection involving the lung or sinonasal tract. Both invasive and noninvasive forms of fungal rhinosinusitis can be associated with calcium oxalate crystal deposition. Here, we report a unique case of sinonasal oxalosis presenting as a destructive lesion in the absence of invasive fungal disease. Due to the clinical and pathologic significance of calcium oxalate crystals as seen in this patient, specimens from the sinonasal tract should be evaluated for the presence of these crystals, which may be a surrogate marker for fungal infection and may also independently cause tissue destruction.

Renal mycetoma presenting with features suggesting renal malignancy: A case report.

INTRODUCTION AND IMPORTANCE: Neoplasms of the Kidney are rarely caused by actinomycetoma or other mycetoma species. Actinomycetoma is a neglected tropical disease which is not uncommon in Sudan. Usually, it is presented as skin and subcutaneous tissue lesions or mass and can affect the bone and other soft tissue. Sites of the lesion are found in lower limbs, upper limbs, head and neck and torso. CASE PRESENTATION: A 55-year-old female presented incidental left renal mass on ultrasound examination from the internal medical department. It is presented as a renal mass mimicking renal cell carcinoma with coexistence with another actinomycetoma brain mass. The histopathology report after nephrectomy confirmed the diagnosis. Patients commenced on anti-actinomycetoma treatment after nephrectomy. CLINICAL DISCUSSION: This is the first reported case in our facility which was diagnosed as a renal actinomycetoma. It was treated by surgical excision and received antibacterial treatments. CONCLUSION: This case demonstrates that renal actinomycetoma can occur in an endemic area even without cutaneous or subcutaneous lesions.