Mycetoma in the Togolese: An Update from a Single-Center Experience.

BACKGROUND: Mycetoma is a chronic inflammatory process caused either by fungi (eumycetoma) or bacteria (actinomycetoma). In this retrospective study, we report epidemiologic and histopathological data of mycetoma observed in the Lome Hospital, Togo in a 25-year period (1992-2016). METHODOLOGY: This is a retrospective study, over a period of 25 years, to analyze epidemiological and etiological findings of mycetomas seen in the single laboratory of pathological anatomy of the Lomé, Togo. RESULTS: A total of 61 cases were retrieved from which only 33 cases were included which where clinically and microbiologically confirmed. The mean age of the patients was 29.7 ± 1.34 and a sex ratio (M/F) of 1.5. The majority of patients were farmers (n = 23 cases; 69.7%). Diagnosed etiologic agents were fungal in 24 cases (72.7%) and actinomycotic cases in 9 cases (27.3%). The fungal mycetomas consisted of Madurella mycetomatis (black grains) and Falcifomispora senegaliensis (black grains). The actinomycotic agents were represented by Actinomadura madurae (white grains), Actinomadurae pelletieri (red grains) and Nocardia sp. (yellow grains). CONCLUSION: This report represents a single-center study which provides epidemiologic and histopathological data of mycetoma cases in Togo.

Transtubular excisional biopsy as a rescue for a non-diagnostic stereotactic needle biopsy-case report and literature review.

Stereotactic needle biopsy, a standard of care for acquiring deep-seated pathology, has limitations and risks in some situations. We present an uncommon case with basal ganglia dematiaceous mycetoma. Due to the firm consistency of the lesion, the initial stereotactic needle biopsy failed to provide a diagnosis. In a second operation, transtubular excisional biopsy was successfully performed to remove the entire mycetoma. We reviewed recent case series of transtubular approaches to deep-seated brain lesions and suggest this method could be a rescue for a non-diagnostic stereotactic needle biopsy and even may be the approach of choice in some cases.

Mycetoma and the environment.

Mycetoma is a chronic, incapacitating, destructive inflammatory disease with many serious damaging impacts. Currently, there is no control or prevention program as many of its epidemiological characteristics, such as the causative organisms' ecological niche, natural habitat, primary reservoir, transmission mode, geographical distribution, incidence, and prevalence, remain unclear. This may be due to a lack of research interest, as mycetoma is still a neglected disease and the scarcity of accurate molecular diagnostic techniques in disease-endemic regions for accurate causative microorganisms identification and mapping. With this background, this study set out to address this knowledge gap by considering the mycetoma environmental occurrence predictors. The medical literature obtained data showed a close association between mycetoma occurrence and its environment. The causative microorganisms are available in the environment in active or dormant forms. Animal dung may be a natural niche and reservoir for these organisms, and thorns may facilitate the subcutaneous inoculation. Some environmental factors, such as the soil type and consistency, temperature, water sources, aridity index, and thorny trees, may be risk factors. The population in endemic areas socioeconomic, hygiene, and health education status are contributory factors for mycetoma. The individual's genetic and immunological backgrounds may determine the disease's susceptibility and resistance. Environmental conditions and personal hygiene improvement are mandatory to reduce disease occurrence. Mycetoma spatial mapping can detect disease cluster areas and then develop public health strategies for early case detection and management to reduce the disease burden. More research interests and facilities are needed to understand disease pathogenesis and appropriate patient management better.

Effect of Mycetoma in Lung Transplant Patients - a Single Center Experience.

BACKGROUND: Mycetomas are uncommon in lung transplant recipients. Prior studies have shown increased mortality associated with mycetoma in lung transplant recipients (LTR). We reviewed our center's experience in outcome of LTRs with pre -or post-transplant mycetoma. METHODS: We retrospectively reviewed electronic health records of LTRs performed at our institute between January 1, 2013 to December 31, 2020. RESULTS: Mycetoma was present in less than 1 percent of LTR patients (7/1086). Mean age at the time of the transplant was 65 years. Idiopathic pulmonary fibrosis (5/7), interstitial lung disease (1/7), and sarcoidosis (1/7) were underlying pulmonary diagnoses. Seventy-one percent (5/7) received single lung transplant and 29% received double lung transplant. Seventy-one percent had negative serum galactomannan vs 29% (2/7) of patients who had positive serum galactomannan (one post and one pre). Fifty-seven percent had positive bronchoalveolar aspergillus galactomannan (23% had negative). A total of 42% (3/7) were found to have mycetoma before transplant and 58% (4/7) had mycetoma post transplant. Chest computed tomography findings in all patients were consistent with mycetoma. CONCLUSIONS: In our cohort of patients, mycetoma was not found to be the primary cause of death if diagnosed pre transplant. Transplant recipients with mycetoma pre transplant did not develop invasive fungal infection or mycetoma post transplant. Careful evaluation of lung transplant candidates with mycetoma is critical. Further studies are needed to determine optimal duration of antifungal therapy and to determine if surgical resection may be needed to manage post-lung transplant mycetoma.

[A rare case of huge aspergilloma developed within a lesion of pulmonary fibrosis secondary with a systemic scleroderma in an immunocompetent patient in Madagascar]. / Un cas rare d'aspergillome volumineux développé au sein d'une lésion de fibrose pulmonaire secondaire à une sclérodermie systémique chez une malade immunocompétente à Madagascar.

We report a rare case of a huge aspergilloma developed within a bronchiectasis due to pulling by a pulmonary fibrosis of systemic scleroderma. The patient is a 58-year-old woman presenting a deterioration of the general state associated with repeating hemoptysis, dyspnea, dysphagia, sclérodactylia, generalized cutaneous sclerosis and Raynaud's phenomenon. There was no antecedent pulmonary tuberculosis. The patient had a pulmonary arterial hypertension complicated by a chronic pulmonary heart at the stage of right cardiac decompensation. Aspergillosis serology was positive and the immunological assessment confirmed scleroderma. The computed tomography showed a huge oblong opacity in a small round bell shape ("signe du grelot", Monad's sign) in the left upper lobe developed within a bronchiectasis, and a bilateral pulmonary fibrosis. Although surgery remains the recommended treatment of an aspergilloma, the management of our patient was medical in front of contra-indication for surgery. The evolution was marked by repeating hemoptysis and stability of the pulmonary lesions 2 years later. The management of this entity remains difficult and complicated; the prognosis is in general unfavourable and depends at the same time on the evolution of scleroderma and the aspergilloma infection.

Pulmonary Madurella mycetomatis mycetoma secondary to knee eumycetoma, Senegal.

Mycetoma is a neglected tropical disease which is endemic in Senegal. Although this subcutaneous mycosis is most commonly found on the foot, extrapodal localisations have also been found, including on the leg, knee, thigh, hand, and arm. To our knowledge, no case of blood-spread eumycetoma has been reported in Senegal. Here, we report a case of pulmonary mycetoma secondary to a Madurella mycetomatis knee eumycetoma. The patient was a 41-year-old farmer living in Louga, Senegal, where the Sudano-Sahelian climate is characterised by a short and unstable rainy season and a steppe vegetation. He suffered a trauma to the right more than 20 years previously and had received treatment for more than 10 years with traditional medicine. He consulted at Le Dantec University Hospital in Dakar for treatment of a right knee mycetoma which had been diagnosed more than 10 years ago. He had experienced a chronic cough for more than a year; tuberculosis documentation was negative. Grains collected from the knee and the sputum isolated M. mycetomatis, confirmed by the rRNA gene ITS regions nucleotide sequence analysis. An amputation above the knee was performed, and antibacterial and antifungal therapy was started with amoxicillin-clavulanic acid and terbinafine. The patient died within a month of his discharge from hospital.

Climate, soil type, and geographic distribution of actinomycetoma cases in Northeast Mexico: A cross-sectional study.

BACKGROUND: Mycetoma is a chronic, granulomatous infection of subcutaneous tissue, that may involve deep structures and bone. It can be caused by bacteria (actinomycetoma) or fungi (eumycetoma). There is an epidemiological association between mycetoma and the environment, including rainfall, temperature and humidity but there are still many knowledge gaps in the identification of the natural habitat of actinomycetes, their primary reservoir, and their precise geographical distribution. Knowing the potential distribution of this infection and its ecological niche in endemic areas is relevant to determine disease management strategies and etiological agent habitat or reservoirs. METHODOLOGY/PRINCIPAL FINDINGS: This was an ambispective descriptive study of 31 patients with actinomycetoma. We determined the biophysical characteristics including temperature, precipitation, soil type, vegetation, etiological agents, and mapped actinomycetoma cases in Northeast Mexico. We identified two disease cluster areas. One in Nuevo Leon, with a predominantly kastanozems soil type, with a mean annual temperature of 22°, and a mean annual precipitation of 585.2 mm. Herein, mycetoma cases were produced by Actinomadura pelletieri, Actinomadura madurae, Nocardia brasiliensis, and Nocardia spp. The second cluster was in San Luis Potosí, where lithosols soil type predominates, with a mean annual temperature of 23.5° and a mean annual precipitation of 635.4 mm. In this area, all the cases were caused by N. brasiliensis. A. madurae cases were identified in rendzinas, kastanozems, vertisols, and lithosols soils, and A. pelletieri cases in xerosols, kastanozems, and rendzinas soils. Previous thorn trauma with Acacia or Prosopis plants was referred by 35.4% of subjects. In these states, the presence of thorny plants, such as Acacia spp., Prosopis spp., Senegalia greggi, Vachellia farnesiana and Vachellia rigidula, are common. CONCLUSIONS/SIGNIFICANCE: Mapping this neglected tropical infection aids in the detection of disease cluster areas, the development of public health strategies for early diagnosis and disease prediction models; this paves the way for more ecological niche etiological agent research.

Difficult mycoses of the skin: advances in the epidemiology and management of eumycetoma, phaeohyphomycosis and chromoblastomycosis.

PURPOSE OF REVIEW: Eumycetoma, phaeohyphomycosis and chromoblastomycosis are subcutaneous mycoses having in common the fact that they are acquired as a result of penetrating trauma to the skin and may be caused by a large variety of fungi. This article will review recent data regarding the epidemiology and treatment of these infections. RECENT FINDINGS: Recent epidemiologic observations in these mycoses include an increased incidence of phaeohyphomycosis in immunosuppressed patients, the association of polymorphisms in genes involved in innate immunity, the occurrence of eumycetoma caused by Madurella mycetomatis and the nosocomial acquisition of phaeohyphomycosis. The management of these infections continues to be challenging. An approach that involves early diagnosis, the use of systemic antifungal agents and local therapies, including surgical removal of lesions, is the basis of the treatment of these diseases. SUMMARY: It is crucial that the epidemiology and clinical presentation of these infections are understood if we are to improve their outcomes.

Cutaneous pseudallescheria boydii infection in a renal transplant patient: A case report.

We describe the case of a 59-year-old male renal transplant recipient who presented with a 1 month history of nodular, erythematous, and crusted lesions on the anterior surface of the left leg. There was no history of trauma. The patient had no systemic signs or symptoms. A skin biopsy revealed evidence of a deep fungal infection, with septate and branching hyphae amongst the dermal inflammatory infiltrate, but the culture was negative. Initial treatment with itraconazole proved ineffective and new lesions appeared. New tissue samples were obtained for culture, allowing the isolation of Pseudallescheria boydii. The patient underwent surgical excision of the lesions combined with voriconazole 400 mg/day for 2 months. After this period no new lesions appeared and the patient has remained without recurrence after 2 years of follow-up. Pseudallescheria boydii is an opportunistic fungus that can cause systemic infection mainly in immunocompromised patients that manifests as pulmonary, osteoarticular, ocular, vascular, cutaneous or central nervous system disease. Resistance to therapy can occur with infection progression and high mortality. Treatment with the combination of an antifungal agent, mainly voriconazole, and surgery, when feasible, probably provide the best results in cutaneous infections.

Keratinocyte infection by Actinomadura madurae triggers an inflammatory response.

BACKGROUND: Actinomycetoma is a syndrome of the skin characterized by chronic inflammation and lesions with nodular grain-like structures. The most common aetiological agents are Nocardia brasiliensis and Actinomadura madurae. In response to infection with these organisms the body produces an inflammatory immune response in the skin. The aim of the present study was to determine the production of chemokines, pro-inflammatory cytokines, antimicrobial peptides and the expression of Toll-like receptors (TLRs) in keratinocytes infected by A. madurae. METHODS: A cell line of HaCaT keratinocytes was infected with A. madurae at a multiplicity of infection of 20:1 for 2 h and the samples were collected from 2 to 72 h post-infection. Intracellular replication of the bacterium was evaluated by counting of colony-forming units, the TLR expression and antimicrobial peptide production were assayed by confocal microscopy and chemokine and pro-inflammatory cytokine levels were determined by enzyme-linked immunosorbent assay. RESULTS: Early in the infection, A. madurae was able to achieve intracellular replication in keratinocytes, however, the cells eventually controlled the infection. In response to the infection, keratinocytes overexpressed TLR2 and TLR6, produced high concentrations of cytokines monocyte chemoattractant protein-1, interleukin 8, human ß-defensin-1, human ß-defensin-2 and LL37 and low levels of tumour necrosis factor α. CONCLUSIONS: The human keratinocytes contribute to the inflammatory process in response to A. madurae infection by overexpressing TLRs and producing chemokines, pro-inflammatory cytokines and antimicrobial peptides.

[Pulmonary Metastase of a Knee Mycetoma in Senegal]. / Métastase pulmonaire d'un mycétome du genou au Sénégal.

Mycetoma is transmitted by thorns infected. The commonest site for mycetoma is the foot. The primary pulmonary are rare and usually secondary to other primary site. We report a case of pulmonary fungal mycetoma secondary to primary site in the knee. We do a review of the literature and we discuss the way of dissemination.

Le mycétome se transmet principalement par piqures d'épines d'arbustes infectés. Les localisations primitives au niveau du pied sont les plus fréquentes. Les localisations pulmonaires sont exceptionnelles et secondaires à des localisations périphériques primitives. Nous rapportons un cas de localisation pulmonaire d'un mycétome fongique secondaire à une localisation au niveau du genou, puis nous faisons une revue de la littérature et nous discutons de la voie de dissémination.

Pseudallescheria boydii brain abscess in a renal transplant recipient: first case report in Southeast Asia.

Pseudallescheria boydii and its asexual form, Scedosporium apiospermum, are ubiquitous filamentous fungi that rarely cause central nervous system (CNS) infection. Brain abscess caused by P. boydii is a highly lethal infection, usually seen in organ transplant recipients who receive a number of immunosuppressive agents. We have presented a case of a 48-year-old man 6 years after renal transplantation who received methylprednisolone followed by antithymocyte globulin for treatment of acute cellular rejection. Eight weeks later, he developed fever, headache, and left-sided hemiparesis. Further investigation with magnetic resonance imaging of the brain showed multiple ring-enhancing hypodense lesions with marked edema which were compatible with brain abscesses. Following surgical drainage, multiple fungal elements were initially described as Aspergillus species. The patient failed to improve and died from rapidly progressive infection despite treatment with amphotericin B. Later a diagnosis was finally made by the isolation of P. boydii in pus culture. The specific diagnosis is difficult to rapidly make, because P. boydii mimics other fungi morphologically in tissue sections and may produce infections clinically similar to other mycoses. Culture of the organism is required for definitive diagnosis. P. boydii infections are important complications of transplantation. They are difficult to treat due to resistance to amphotericin B. Physicians should consider P. boydii a possible cause of brain abscess in organ transplant recipients, especially with heavy immunosuppressive agents. This is the first case report of CNS infection due to P. boydii in a renal transplant patient in Southeast Asia.

Fungal vertebral osteomyelitis due to Scedosporium apiospermum after near-drowning.

Near-drowning predisposes one to infection by the fungus Scedosporium apiospermum, and brain abscess is the most common consequence. Vertebral osteomyelitis due to this organism is exceedingly rare. The authors report on a 43-year-old man who developed fungal spondylodiscitis several weeks after a near-drowning event. The rare nature of this infection led to a delay in diagnosis. A combination of surgical debridement, instrumentation, and antifungal therapy resulted in an excellent outcome at 1 year of follow-up.

Late complication after tropic storm accident: subcutaneous and intracranial actinomycetoma.

We report a 53-year-old farmer who developed subcutaneous and cerebral masses 24 years after penetrating trauma during a tropic storm. Computed tomography scans, magnetic resonance imaging and histopathology disclosed actinomycetoma, a disease that rarely develops after trauma and is only occasionally seen with intracranial manifestation. Clinically, the cutaneous manifestation resembled acne keloidalis nuchae or dissecting folliculitis of the scalp. He was treated by neurosurgery and antibiosis.

Mycetomas: an epidemiological, etiological, clinical, laboratory and therapeutic review.

Mycetoma is a chronic suppurative disease of the skin and subcutaneous tissue, characterized by a symptomatic triad: tumor, fistulas and grains. It can be caused by fungi (eumycetoma) and bacteria (actinomycetoma), with similar clinical features. Diagnosis is based on the clinical presentation and identification of the etiological agents in the tissue, by mycological/bacteriological, histopathological and immunohistochemical tests. It is important to specify the fungal or bacterial etiology, because the treatments are different. An approach that involves early diagnosis, the use of systemic antibiotics or antifungal agents, including surgical removal of lesions, is the basis for the treatment of these diseases. In this review, the most commonly used diagnostic methods and treatments will be discussed. Also, we will review the history of the disease through epidemiological and etiological aspects.

Disseminated Scedosporium apiospermum infection in renal transplant recipient: long-term successful treatment with voriconazole: a case report.

Scedosporium apiospermum, the asexual form of Pseudallescheria boydii, is a ubiquitous fungus that represents an unfrequent complication of immune suppression. It accounts for 20% of all non-Aspergillus mold infections in organ transplant recipients. The infection can be localized or disseminated in multiple organs, including lungs, brain, joints, tendons, and skin, and is difficult to treat, due to resistance of S apiospermum to amphotericin B and other antifungal agents. The mortality rate is about 50%. To our knowledge, there are no prospective studies or registries of transplant recipients to guide diagnosis and there are no evidence-based recommendations for the optimal management of this infection. We report a case of S apiospermum infection in a woman with renal transplantation. The first occurrence of infection was a solitary nodule on the forearm, which was surgically excised. Two following relapses were disseminated to the knee, the Achilles tendon, and the skin of the left leg. The infection was successfully treated with voriconazole, but due to the severe iatrogenic immune suppression, a strong reduction in immunosuppressant drugs was needed.

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)