A Case Report of a Mysterious Mycetoma.

Mycetoma, a chronic subcutaneous infection caused by bacterial or fungal species from soil and water, presents a diagnostic challenge due to its rarity and diverse clinical manifestations. Predominantly affecting male workers in endemic regions, mycetoma typically manifests as painless swelling evolving into purulent lesions with draining sinuses in the extremities. Although historically uncommon in regions like North America, rising immigration and international travel have led to an increased prevalence, necessitating heightened clinical suspicion. Early diagnosis is crucial to prevent severe complications such as limb loss and septicemia. This case report details the diagnosis and management of chronic actinomycetoma due to Nocardia spp. in a Guatemalan immigrant landscaper and emphasizes the importance of comprehensive understanding and timely intervention in mycetoma cases.

Naphthylisoquinoline alkaloids: novel agents against the causative pathogens of eumycetoma and actinomycetoma-en route to broad-spectrum antimycetomal drugs.

Mycetoma is a devastating neglected tropical infection of the subcutaneous tissues. It is caused by fungal and bacterial pathogens recognized as eumycetoma and actinomycetoma, respectively. Mycetoma treatment involves diagnosing the causative microorganism as a prerequisite to prescribing a proper medication. Current therapy of fungal eumycetoma causative agents, such as Madurella mycetomatis, consists of long-term antifungal medication with itraconazole followed by surgery, yet with usually unsatisfactory clinical outcomes. Actinomycetoma, on the contrary, usually responds to treatment with co-trimoxazole and amikacin. Therefore, there is a pressing need to discover novel broad-spectrum antimicrobial agents to circumvent the time-consuming and costly diagnosis. Using the resazurin assay, a series of 23 naphthylisoquinoline (NIQ) alkaloids and related naphthoquinones were subjected to in vitro screening against two fungal strains of M. mycetomatis and three bacterial strains of Actinomadura madurae and A. syzygii. Seven NIQs, mostly dimers, showed promising in vitro activities against at least one strain of the mycetoma-causative pathogens, while the naphthoquinones did not show any activity. A synthetic NIQ dimer, 8,8'''-O,O-dimethylmichellamine A (18), inhibited all tested fungal and bacterial strains (IC50 = 2.81-12.07 µg/mL). One of the dimeric NIQs, michellamine B (14), inhibited a strain of M. mycetomatis and significantly enhanced the survival rate of Galleria mellonella larvae infected with M. mycetomatis at concentrations of 1 and 4 µg/mL, without being toxic to the uninfected larvae. As a result, broad-spectrum dimeric NIQs like 14 and 18 with antimicrobial activity are considered hit compounds that could be worth further optimization to develop novel lead antimycetomal agents.

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.

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.

Madura foot and a continued diagnostic enigma: Dot-in-circle sign on magnetic resonance imaging and ultrasound.

Madura foot is a chronic granulomatous disease of the skin and underlying tissues caused by fungi or bacteria. Early diagnosis is important to avoid disfiguring limb deformities. Low clinical suspicion, limited availability of diagnostic tools, and infection with fastidious organisms may lead to misdiagnosis and delayed treatment. Imaging tests can help to make a timely diagnosis in a non-invasive manner. Here we report two patients with a non-classical clinical presentation and a more favorable differential diagnoses who were correctly diagnosed as cases of Madura foot based on the ''dot-in-circle sign'', a specific finding on magnetic resonance imaging and ultrasound.

Actinomycetoma distal radius mimic osteosarcoma: A case report of a rare presentation.

Key Clinical Message: Actinmomycetma is a granulomatous infection with a presentation was very similar to osteosarccoma. Multidisciplinary team and triple assessments are extremely important to prevent misdiagnosis, surgical treatment in combination with medical treatment followed by regular clinical and radiological follow-up can be a limb-saving procedure in such cases. Abstract: Various conditions may mimic osteosarcoma. The differential diagnosis of osteosarcoma is broad; Includes tumors, infection, trauma, and inflammatory processes arising from the musculoskeletal system. A proper history, examination, diagnostic imaging studies, and pathological analysis are essential to establish a precise diagnosis. This case report serves to illustrate the significance of recognizing the similarities between these two lesions and other rare features that will help to differentiate between actinomycetoma and osteosarcoma, to prevent late or misdiagnosis.

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.

Mycetoma (Madura foot): A Case Report of a Rare Tropical Disease in Turkey.

Mycetoma is a chronic discharging infection involving the skin, subcutaneous tissue, fascia, and bone, which is endemic in tropical and subtropical countries. We report a rare case of mycetoma localized on the foot of a patient living in a country with a temperate climate. A 32-year-old male patient presented with painless swelling in the right foot. He had undergone surgery 3 years ago with the same complaints. Magnetic resonance imaging revealed a 90 × 65 × 37 mm cystic soft tissue lesion in the posterior of the right ankle. Histological analysis identified the fungus. Extensive resection was performed. Mycetoma is characterized by chronic granulomatous inflammatory response, often associated with sinus tract formations due to fungal (eumycetoma) or bacterial (actinomycetoma) organisms. Here we provide a literature review and highlight the importance of increasing awareness toward mycetoma, particularly in non-endemic regions.

A scoping review of mycetoma profile in Egypt: revisiting the global endemicity map.

Mycetoma is a chronic infectious disease endemic in sub-Saharan Africa (SSA), India and parts of South and North America. The epidemiologic profile of the disease in Egypt, which neighbours SSA, has not been explored previously. Therefore we conducted a scoping review of the literature on mycetoma in Egypt. We searched the literature comprehensively on MEDLINE and Google Scholar using free-text words and Medical Subject Headings and terms. Both published and non-peer-reviewed (grey literature) articles were included. The initial search identified 133 reports. Of these, only eight were found to be relevant and were included in the study. The total number of mycetoma patients was 59, reported between 1949 and 2015. There was a predilection for eumycetoma (44 of 59) patients (75%), while actinomycetoma constituted 15 patients (25%). Six patients were female, 28 were male and 25 were unreported. Children and adolescents constituted 3 of 59 (5%), 52 (88%) were adults and age was not provided for 4 patients. Only four patients (7%) were non-autochthonous. The incidence of mycetoma in Egypt is higher than previously reported. Egypt is probably a low-endemic country. An accurate estimate of the prevalence and epidemiology of mycetoma necessitates further research collaboration.

Genetic variability of the 16S rRNA gene of Nocardia brasiliensis, the most common causative agent of actinomycetoma in Latin America and the Caribbean

ABSTRACT Mycetoma is a neglected tropical disease (NTD) declared by the World Health Organization (WHO) in 2016. It is characterized by the progressive growth of nodules and granulomatous lesions on the legs, arms, and trunk. It is potentially disfiguring and causes disability or amputations in working-age people from marginalized areas. The causative agents can be fungi (eumycetoma) or actinobacteria (actinomycetoma), the latter being the most common in America and Asia. Nocardia brasiliensis is the most important causal agent of actinomycetoma in the Americas. Taxonomic problems have been reported when identifying this species, so this study aimed to detect the 16S rRNA gene variations in N. brasiliensis strains using an in silico enzymatic restriction technique. The study included strains from clinical cases of actinomycetoma in Mexico, isolated from humans and previously identified as N. brasiliensis by traditional methods. The strains were characterized microscopically and macroscopically, then subjected to DNA extraction and amplification of the 16S rRNA gene by PCR. The amplification products were sequenced, and consensus sequences were constructed and used for genetic identification and in silico restriction enzyme analysis with the New England BioLabs® NEBcutter program. All study strains were molecularly identified as N. brasiliensis; however, in silico restriction analysis detected a diversity in the restriction patterns that were finally grouped and subclassified into 7 ribotypes. This finding confirms the existence of subgroups within N. brasiliensis. The results support the need to consider N. brasiliensis as a complex species.

Case report: Mycetoma caused by Gordonia soli

ABSTRACT Mycetoma is a neglected tropical disease caused by fungi (eumycetoma) or bacteria (actinomycetoma), with high morbidity. Gordonia spp. are gram-positive bacteria that have previously been reported to cause mycetoma. Here, we report a case of Gordonia soli (initially misidentified as Nocardia spp.) as the etiological agent of actinomycetoma in a 64-year-old patient. After a literature search in the Cochrane Library, LILACS, SciELO, MEDLINE, PubMed, and PubMed Central databases, we concluded that this is the first case report of mycetoma caused by Gordonia soli. The current case highlights the importance of microbiological diagnosis of mycetoma and the challenges in its management.

Histopathological Diagnosis of Eumycetoma With Paraspinal Sinuses: A Rare Case Report.

Mycetoma, well known as "Madura foot," is a long-standing granulomatous infection of the skin and subcutaneous tissue. Causative organisms are filamentous fungi (eumycetes) and bacteria (actinomycetes). It characteristically presents with firm woody swelling, discharging sinuses with grains (containing the causative organism). Diagnosis in suspected cases can be made by microbiological evaluation, histopathological, cytological and radio imaging techniques. To differentiate between eumycetoma and actinomycetes histopathology is an accurate diagnostic modality as seen in the present case. We report a case of 42 years male who presented with swelling on his back with discharging sinus. Histopathological specimen containing multiple, irregular, greyish, whitish tissue pieces with skin attached all together measuring around 12×9×4 cm from the paraspinal region. The section shows histopathological features suggestive of eumycetoma. Periodic acid -Schiff stain showed the presence of septate, branching fungal hyphae and black granules. Eumycetoma can be accurately diagnosed by histopathological evaluation using a special stain. It is confirmatory and provides a guide for treatment plans with a high index of suspicion.

Diagnostics to support mycetoma management-Development of two target product profiles.

OBJECTIVE: Mycetoma is a neglected tropical disease caused by more than 70 different microorganisms and identified by the WHO as one of the high-priority diseases for developing diagnostic tests. To ensure the production of diagnostic assays for use by clinical staff in endemic regions, target product profiles (TPPs) were designed. METHODS: We describe the development of two TPPs: one for a diagnostic test able to identify the causative agent of mycetoma and another that would determine when treatment could be stopped. The TPPs were developed by considering product use, design, performance, product configuration and costs. RESULTS: Version 1.0 TPPs for two uses were posted by WHO for a 1-month online public consultation on 25 October 2021, and the final TPP was posted online on 5 May 2022. CONCLUSION: A major difficulty encountered in developing both TPPs was the large number of agents able to cause mycetoma and the lack of specific biomarkers for most of them.

Staphylococcus aureus causing primary foot botryomycosis mimicking actinomycetoma: a case report from Sudan.

OBJECTIVES: Botryomycosis is a rare chronic granulomatous inflammatory disease of bacterial origin. Two forms of the disease exist; the cutaneous and the visceral form. The subcutaneous form mimics actinomycetoma clinically and histologically; however, the treatment is different. In this communication, we report on a Sudanese male patient who presented with foot botryomycosis. DESIGN: Case report. RESULTS: The patient was initially diagnosed with actinomycetoma by the presence of Streptomyces somaliensis like-grains in the histological slides. The patient was treated with a combination of co-trimoxazole and amikacin sulfate and shifted after 1 year to co-trimoxazole, amoxicillin, and clavulanic acid. Despite treatment, the infection progressed, and the bone was invaded. The infected limb was amputated. The histopathological report of the surgical biopsy showed gram-positive cocci inside the grain. The 16S sequence identified these cocci as Staphylococcus aureus. CONCLUSION: This is the first reported botryomycosis case from Sudan, and it highlights why molecular identification is vital in diagnosis.

Micetoma por Nocardia asteroides / Nocardia Asteroids Mycetoma

Introducción: En 1860 Henry Van Carter introdujo la definición del micetoma y desde 1884 se reportan los primeros casos en África (Sudán, Senegal). Estas infecciones afectan la piel, el tejido celular subcutáneo y, en ocasiones, los músculos, los huesos, y pueden diseminarse por la cavidad torácica, la abdominal, y por otras regiones del cuerpo. Objetivo: Presentar un caso de micetoma por Nocardia asteroides con evolución desfavorable y tratamiento. Presentación del caso: Paciente masculino de 47 años de edad que sufrió hace 18 años un trauma en la rodilla izquierda con herida avulsiva y evolución desfavorable por infección. Diez años después presentó en el mismo sitio múltiples lesiones fistulosas con drenaje activo, secreción serohemática escasa y no fétida. En varias ocasiones fue llevado al salón de operaciones para realizarle debridamientos quirúrgicos y toillete y recibió múltiples tratamientos antibióticos y antifúngicos. Se concluyó el caso como un micetoma y se aisló una Nocardia asteroides. El paciente estuvo en desacuerdo con la amputación de la extremidad como tratamiento quirúrgico definitivo. Llegó a nuestro centro en octubre del 2020 con mal estado general y extensión severa del proceso infeccioso en toda la extremidad. Se planificó una hemipelvectomía como tratamiento definitivo, pero desafortunadamente el paciente falleció antes, debido a complicaciones generales. Conclusiones: Ante la aparición del micetoma es importante definir el alcance de la infección para determinar el tipo de tratamiento a utilizar, ya que bien empleado y de forma oportuna, puede salvar la vida al paciente sin dejar graves secuelas(AU)

Introduction: In 1860, Henry Van Carter introduced the definition of mycetoma and since 1884 the first cases have been reported in Africa (Sudan, Senegal). These infections affect the skin, the subcutaneous cellular tissue and, sometimes, the muscles, the bones, and it can spread throughout the thoracic cavity, the abdominal cavity, and other regions of the body. Objective: To report a case of mycetoma due to nocardia asteroides with unfavorable evolution and treatment. Case report: We report the case of a 47-year-old male patient who suffered a left knee trauma 18 years ago with an avulsive wound and unfavorable evolution due to infection. Ten years later, he presented, in the same site, multiple fistulous lesions with active drainage, scant serohematic, non-fetid secretion. On several occasions he was taken to the operating room for surgical debridement and toilette and he received multiple antibiotic and antifungal treatments. The case was concluded as a mycetoma. Nocardia asteroides was isolated. The patient disagreed with limb amputation as definitive surgical treatment. He came to our treatment center in October 2020 with poor general condition and severe extension of the infectious process throughout the limb. A hemipelvectomy was planned as definitive treatment, but unfortunately the patient deceased before due to general complications. Conclusions: Before the appearance of mycetoma, it is important to define the extent of the infection to determine the type of treatment to use, since it can save the patient's life if properly used and in a timely manner without leaving serious sequelae(AU)

Diagnostic imaging of foot mycetomas: A report on two cases.

Mycetomas caused by aerobic actinomycetes are called actinomycetomas. It is primarily localized in subcutaneous tissue but it can spread to different tissue planes including the skin, deep tissues and structures and eventually the bones. We report the cases of 2 patients referred for evaluation of soft tissue masses involving the foot. A 40-year-old male and a 25-year-old male, in both cases MRI was performed to assess the extension, which was later completed by a CT scan. MRI revealed a low intensity matrix that represents fibrosis containing multiple high intensity lesions corresponding to the mycetoma grains. Within some of the lesions a low-intensity focus was identified. This "dot-in-circle sign" on an MRI is a pathognomonic feature of mycetoma. The purpose of this work is to describe the characteristic MRI appearance of foot mycetoma.

Mycetoma in north-western Yemen: Clinico-epidemiological and histopathological study.

Background Mycetoma is widespread in Yemen; however, there are only a few documented reports on the entity from this geographical area. Methods A prospective study of 184 cases of mycetoma (male 145 and female 39) from different regions of north-western Yemen was conducted between July 2000 and May 2014. Clinical profile was recorded in a standardized protocol. The diagnosis was based on clinical features, X-ray studies, examination of grains, and histopathology. Results Eumycetoma was diagnosed in 129, caused by Madurella mycetomatis in 124, Leptosphaeria senegalensis in one and pale grain fungus in four, whereas actinomycetoma occurred in 55, caused by Streptomyces somaliensis in 29, Actinomadura madurai in nine, Actinomadura pelletieri in one, and Nocardia in sixteen. Eumycetoma cases were treated with prolonged course of antifungal drugs, mostly ketoconazole, with itraconazole being used in four patients, along with excision or debulking. Results were better when antifungal drugs were given two to three months before surgery and in those who received itraconazole. Actinomycetoma cases were initially treated with co-trimoxazole monotherapy; later streptomycin was added in 30 cases. Six patients who did not show adequate improvement and two others from the start were treated with modified Welsh regimen and with good results. Limitations Identification of different causative agents was done by histopathology and could not be reconfirmed by culture. Conclusion Mycetoma is widespread in north-western Yemen with a higher incidence of eumycetoma and a majority of the cases were caused by Madurella mycetomatis. Modified Welsh regimen in actinomycetoma and itraconazole with excision in eumycetoma showed the best results.

Orbital Actinomycetoma with cranial extension: A rare case report.

INTRODUCTION: Mycetoma is a neglected tropical disease that commonly affects the lower extremity. The disease is attributed to subcutaneous granulomatous inflammation leading to distinct clinical features of gradual painless swelling accompanied by nodules and drains. Orbital mycetoma is an extremely rare entity of the disease. We reported the clinical presentation, diagnosis, and surgical outcomes in a case of orbital mycetoma with cranial extension. CASE PRESENTATION: A 25-years-old male complained of left eye protrusion for 8 years, followed by complete loss of vision on the left eye for 7 years and eventually left eye pain for the last year. The left eye was displaced anteriorly and inferiorly with normal oculomotor, abducent, and trochlear examination. Brain CT scan showed an increase in orbital bone thickness with extension to the anterior cranial base, middle cranial base, and the orbital process of the zygomatic bone. MRI revealed a large lesion involving the left frontotemporal region with extension to left orbit, left posterior ethmoid air cells, and left temporal suprasellar region. The lesion was homogenously enhanced with contrast. The patient underwent a left orbitozygomatic cranitomy for resection of the lesion. However, total resection was inapplicable due to the extension of the bony lesion up to the petrous bone. Cranioplasty was performed by titanium mesh. CONCLUSION: Mycetoma is a chronic inflammatory disease affecting subcutaneous tissues commonly in the lower limbs. The disease can be caused by fungi (Eumycetoma) or bacteria (Actinomycetoma). Orbital mycetoma is an extremely rare entity of the disease. However, it is commonly associated with the cranial extension. Early diagnosis and prompt surgical and medical treatment are the keys to good outcomes.

Spinal and cranio-cervical mycetoma: A difficult surgery, with poor prognosis.

BACKGROUND: Few central nervous systems (CNS) cases of actinomycetoma have been recorded in the literature, and most were reported in tropical and subtropical regions. The management of this invasive infection is difficult, especially when it affects the spine and the cranio-cervical regions. CASE: We report an unusual case of a cranio-cervical junction actinomycetoma, in a patient presenting a cerebellar syndrome from brainstem compression. The CT scan showed a compressive solid osteolytic lesion in the cranio-cervical junction. The patient underwent cranio-cervical decompression and lesion resection. The diagnosis of actinomycetoma was confirmed on immune-histochemistry and molecular analysis. At 4 months' follow-up, the patient presented a fatal recurrence disseminating within the cerebellum and the spine. CONCLUSION: The surgical treatment of CNS actinomycetoma presented poor prognosis and a disseminating recurrence. We believe that clinicians and surgeons must be informed about these "new" infectious pathologies that are so difficult to treat, especially with the arrival of migrant patients from endemic countries in conflict.