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.

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.

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.

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)

Alleviating Diagnostic Dilemma of Maduramycosis: A Case Series

@#Maduramycosis1 is chronic infection of cutaneous and subcutaneous tissue caused by bacteria and fungi. It involves skin, subcutaneous tissue and bones.2 Here we report a case series of 14 patients of mycetoma describing their epidemio-clinical features and laboratory investigations. The most common clinical presentation in the patients were infiltrated subcutaneous swelling with multiple discharging sinus tracts (fistulas). Lesions were located on the foot in all the cases.

Actinomicetoma plantar en un paciente sin factores de riesgo / Plantar actinomycetoma in a patient without risk factors

Resumen Se presenta un caso clínico de un actinomicetoma plantar en un paciente sin factores de riesgo, cuyo diagnóstico fue realizado mediante una biopsia de tejido plantar por sospecha de una neoplasia. Dado que el paciente no respondió satisfactoriamente a la terapia de primera línea, debió completar 24 semanas de tratamiento con doxiciclina, a lo cual evolucionó favorablemente. Finalmente, se desarrolla una breve discusión sobre los micetomas plantares.

A case of plantar actinomycetoma without risk factors is presented, which was diagnosed by hystopatological analysis of a foot biopsy because of the suspicion of neoplasia. Since the patient did not fully respond to the first-line therapy antibiotics, a 24-weeks doxycycline regime was started, achieving a satisfactory response. Finally, a brief discussion on plantar mycetomas is presented.

Actinomicetoma torácico con afección pulmonar. Tratamiento con carbapenémico y aminoglucósido / Thoracic actinomycetoma with lung compromise. Treatment with carbapenem and aminoglycoside

Resumen: El actinomicetoma, infección crónica, granulomatosa y progresiva de la piel y tejido celular subcutáneo que afecta más las extremidades inferiores y en especial los pies, se caracteriza por la tríada de trayectos fistulosos, aumento de volumen y granos. La afección en el tórax en México ocupa el segundo lugar en cuanto a frecuencia. Sin embargo, el daño pulmonar es muy raro, afecta menos de 1% de los casos, éste puede ocurrir por continuidad al afectar el tronco, lo que causa tratamientos antimicrobianos prolongados con falta de apego a los mismos, así como aumento de la mortalidad. El objetivo de este artículo es comunicar un caso clínico de actinomicetoma pulmonar, así como la respuesta al tratamiento prescrito a este paciente con base en carbapenémicos, aminoglucósidos y trimetoprim con sulfametoxazol.

Abstract: Actinomycetoma is a chronic, granulomatous and progressive infection of the skin and subcutaneous cellular tissue that affects the lower limbs and especially the feet, characterized by triad of localized swelling, draining sinuses and grains. The chest condition in Mexico ranks second in frequency. Nevertheless, pulmonary affectation is very rare with less than 1%, this can occur by continuity affecting the trunk leading to prolonged antimicrobial therapy and lack of attachment to it, as well as increased mortality. The aim of this paper is to report a case of pulmonary actinomycetoma as well as response to treatment with carbapenems, aminoglycoside and trimethoprim with sulfamethoxazole.

Actinomicetoma en el embarazo. Reporte de un caso: ¿Qué hacer y cómo tratarlo? / Actinomycetoma in pregnancy. Case report: what to do, how to handle?

Resumen ANTECEDENTES: El micetoma es una infección causada por hongos y actinomicetos aeróbicos. Es un padecimiento frecuente en México, con mayor afectación en hombres que en mujeres (3:1). Se caracteriza por aumento de volumen, deformidad del sistio de lesión y formación de fístulas. OBJETIVO: Reportar un caso de actinomicetoma durante el embarazo y analizar el protocolo de tratamiento. CASO CLINICO: Paciente de 29 años de edad, en su cuarto embarazo, con 29 semanas de gestación. El padecimiento se inició 13 años atrás, con dermatosis localizada en la extremidad inferior izquierda, constituida por un nódulo indoloro, que permaneció sin cambios hasta el inicio de la gestación actual, cuando le aparecieron múltiples lesiones nodulares y fístulas. En el examen directo de la secreción se observaron granos y en el cultivo se identificó Nocardia brasiliensis. Después del embarazo a término y con recién nacido sano, si indicó lactancia durante 4 meses y se interrumpió para prescribir bromocriptina. La dermatosis se extendió al doble, sin afectación ósea; se indicó tratamiento con sulfametoxazol-trimetoprima y dapsona. El tiempo total de tratamiento fue de 15 meses y seguimiento sin medicación durante un año. Se obtuvo curación clínica y microbiológica. CONCLUSIONES: El micetoma en mujeres embarazadas es excepcional. La mayor parte de los portocolos de tratamiento deben contraindicarse durante el embarazo. Cuando el micetoma se localiza en una zona que no afecta otros órganos ni se extiende, se sugiere continuar el embarazo y la lactancia sin prescripción de medicamentos pero iniciarla posterior al nacimiento.

Abstract BACKGROUND: Mycetoma is an infection caused by fungi and aerobic actinomycetes. It is a frequent condition in Mexico; it presents less in women than men (1:3). It is characterized by increased volume deformity of the region and sinuses. OBJECTIVE: We present a case of actinomycetoma in a pregnant patient and to analyze the behavior in its therapeutic management CLINICAL CASE: We present female, 29 years old, attending her fourth pregnancy at 29 weeks of gestation. It began 13 years ago with a localized dermatosis of the lower left limb, constituted by a painless nodule, remained unchanged until the beginning of the current gestation, developed multiple nodules and sinuses. A direct examination of the secretion was performed, observing grains, Nocardia brasiliensis was identified. After product birth, lactation was allowed for 4 months and discontinued with bromocriptine. The dermatosis extended to double without bone affection, treatment with sulfamethoxazole/trimethoprim + dapsone was given. Total time was 15 months and follow-up without medication for one year. Clinical and microbiological cure was achieved. CONCLUSIONS: The development of mycetoma in pregnant women is rare, it is important to know the etiology, in eumycetoma all the antimycotics are teratogenic and in actinomycetoma most antibiotics cannot be used in pregnancy with some exceptions. If mycetoma is located in an area that does not compromise other organs or does not spread it is best to leave the course of pregnancy and lactation and then start treatment.

Activation and IL-1β secretion of human peripheral phagocytes infected with Actinomadura madurae, Nocardia asteroides and Candida albicans

OBJECTIVE@#To evaluate the ability of Actinomadura madurae (A. madurae) and Nocardia asteroides (N. asteroides), using Candida albicans (C. albicans) as prototypic control, to elicit the activation and IL-1β secretion of blood phagocytic cells from healthy donors.@*METHODS@#Microscopic evaluation of phagocytosis/activation, cell viability and spectrophotometric quantitation of endocytosis/activation, were assessed by using formazan blue test in human blood phagocytes infected with C. albicans, A. madurae or N. asteroides treated with either normal human serum (NHS) or with decomplemented NHS. Interlukin-1β from culture supernatants of infected polymorphonuclear was tested by ELISA kit assay.@*RESULTS@#Microscopic assay showed that phagocytosis and activation of adherent mononuclear phagocytes were greater with C. albicans followed by A. madurae and then by N. asteroides. Spectrophotometric assay in polymorphonuclear phagocytes infected with NHS-treated pathogens indicated that activation was similarly higher by C. albicans and A. madurae and lower by N. asteroides. Kinetic assays in infected polymorphonuclear cells showed that viability was decreased by C. albicans and N. asteroides or unaffected with A. madurae. Levels of IL-1β at 8 h of incubation were higher with C. albicans followed by A. madurae whereas lower levels were found with N. asteroides.@*CONCLUSIONS@#The extent of cell-viability and activation as well IL-1β secretion may be related with the virulence of C. albicans and N. asteroides and other parameters remain to be explored for assessing the virulence of A. madurae.

Activation and IL-1β secretion of human peripheral phagocytes infected with Actinomadura madurae, Nocardia asteroides and Candida albicans

Objective To evaluate the ability of Actinomadura madurae (A. madurae) and Nocardia asteroides (N. asteroides), using Candida albicans (C. albicans) as prototypic control, to elicit the activation and IL-1β secretion of blood phagocytic cells from healthy donors. Methods Microscopic evaluation of phagocytosis/activation, cell viability and spectrophotometric quantitation of endocytosis/activation, were assessed by using formazan blue test in human blood phagocytes infected with C. albicans, A. madurae or N. asteroides treated with either normal human serum (NHS) or with decomplemented NHS. Interlukin-1β from culture supernatants of infected polymorphonuclear was tested by ELISA kit assay. Results Microscopic assay showed that phagocytosis and activation of adherent mononuclear phagocytes were greater with C. albicans followed by A. madurae and then by N. asteroides. Spectrophotometric assay in polymorphonuclear phagocytes infected with NHS-treated pathogens indicated that activation was similarly higher by C. albicans and A. madurae and lower by N. asteroides. Kinetic assays in infected polymorphonuclear cells showed that viability was decreased by C. albicans and N. asteroides or unaffected with A. madurae. Levels of IL-1β at 8 h of incubation were higher with C. albicans followed by A. madurae whereas lower levels were found with N. asteroides. Conclusions The extent of cell-viability and activation as well IL-1β secretion may be related with the virulence of C. albicans and N. asteroides and other parameters remain to be explored for assessing the virulence of A. madurae.

Mycetoma at a tertiary care hospital in Saudi Arabia: Correlation of histopathological and clinical findings

Objective: To present the histopathological and clinical correlation of mycetoma among patients attending King Abdulaziz University Hospital between 1998-2013. Methods: The data of all histopathologically diagnosed mycetomas in the period between January 1998 and January 2013 were collected through a computerized database search of the anatomic pathology archives at King Abdulaziz University Hospital. The collected data were analysed. Identification of species were performed for five patients using 16S ribosomal DNA and internal transcribed spacer 2. Results: There were 19 patients with mycetoma with an average age of 44.26 years and male: female ratio of 4:1. Actinomycetoma were 63.15% and eumycetoma were 36.84%. All patients presented with the classic lesions; presenting as painless subcutaneous mass, sinuses and discharge containing grains. The swellings were of slow evolution, with preferential foot localization. Species specification performed for samples from five patients with active lesions revealed species of Actinomyces israelii and Madurella mycetomatis in respective cases. Conclusions: Actinomycetoma is more common than eumycetoma in this region. The fact that one of the patients with eumycetoma was a Saudi national raises the possibility of an indigenous species similar to Maduraella mycetomatis to be further explored for characteristics and pathogenesis. The disease has to be prioritized again and more robust and quick molecular diagnostic tools should be made available in order to save patients form disfiguring amputations.

Disseminated orbital actinomycetoma: A case report.

Mycetoma is a chronic granulomatous infection. Lower extremities are commonly involved. A 20-year-old male came with complaints of multiple sinuses on scalp, left eyelid swelling with a sinus and dystopia, since one year. On examination there was relative proptosis in left eye of 2 mm. Computed tomography scan showed soft tissue swelling of the pre-septal area of the left upper eyelid with orbital involvement. Magnetic resonance imaging showed increased left orbital volume and evident dystopia. Microbiology testing of the erosive scalp and lid lesions showed genus Nocardia, suggestive of actinomycetoma. This case is presented as it shows an unusual involvement of the orbit.

Micetoma: [revisión] / Mycetoma: [review]

El micetoma es una enfermedad crónica, inflamatoria, subcutánea y granulomatosa, causada por diferentes especies de hongos (eumicetoma), actinomicetos (actinomicetoma) o bacterias como Staphylococcus aureus, Pseudomonas aeruginosa (Botriomicosis). Esta patología ocurre entre latitudes15 al sur y 30 Norte y es endémica en áreas relativamente áridas. Los microorganismos causales están presentes en el suelo y pueden entrar al tejido subcutáneo por inoculación traumática. El micetoma comúnmente afecta adultos entre 20 y 40 años, predominantemente hombres. El pie es el sitio más comúnmente afectado. El micetoma se presenta como una inflamación subcutánea progresiva. Se desarrollan múltiples nódulos, los cuales supuran y drenan a través de senos, descargando granos durante la fase activa de la enfermedad. El diagnóstico se basa en radiología, imágenes ultrasónicas, citología, cultivo e inmunodiagnóstico. El actinomicetoma es susceptible a tratamiento antibiótico prolongado, preferiblemente con varias medicaciones. El Eumicetoma se trata con excisión quirúrgica agresiva combinada con tratamiento médico y la botriomicosis se trata con antibióticos una vez establecido el diagnóstico, ojalá con aislamiento del agente causal.

Mycetoma is a chronic, granulomatous, subcutaneous, inflammatory disease caused by different fangal species (Eumycetoma), actinomycetes (Actimonycetoma), or bacteria Staphylococcus aureus, Pseudomonas aeruginosa, (Botryomycosis). It occurs between the latitudes of 15 South and 30. North and is endemic in relatively arid areas. The organisms are present in the soil and may enter the subcutaneous tissue by traumatic inoculation. Mycetoma commonly affects adults aged 20 to 40 years, predominantly males. The foot is most commonly affected. Mycetoma presents itself as progressive, subcutaneous swelling. Multiple nodules develop which may supúrate and drain through sinuses, discharging grains during the active phase of the disease. Diagnosis may involve radiology, ultrasonic imaging, cytology, culture, histology, or immunodiagnosis. Actinomycetoma is amenable to prolonged treatment by antibiotics, preferably by combining several medications. Eumycetoma is usually treated by aggressive surgical excision combined with medical treatment and Botryomycosis is treated with antibiotics upon establushment of the diagnosis preferably after isolation of the causal agent.

Inmunoglobulinas en pacientes con actinomicetoma por Nocardia brasiliensis / Immunoglobulins in patients with Nocardia brasiliensis actinomycetoma

Considerando que algunos autores han reportado un aumento en la cantidad de algunas inmunoglobulinas en los pacientes con actinomicetoma, en este trabajo nos propusimos determinar diferencias en la producción de IgG1, IgG2, IgG3, IgG4 e IgM en 25 pacientes con actinomicetoma por Nocardia brasiliensis y 25 personas sanas provenientes de una zona endémica de micetoma. La determinación de inmunoglobulinas se realizó por medio de la técnica de ELISA. Para sensibilizar las placas se emplearon 6 antígenos de N. brasiliensis: un antígeno crudo denominado NB y cinco derivados del mismo (NB2, NB4, NB6, NB8 y NB10) separados por punto isoeléctrico. Los niveles de las cuatro subclases de IgG fueron mayores en los sueros de los pacientes que en el suero de los controles, con una diferencia máxima en IgG3 e IgG4; para esta última subclase, los seis antígenos fueron altamente reactivos. La concentración de IgM fue igual en ambos grupos. Es probable que como ocurre en otras infecciones, en la fisiopatogenia del actinomicetoma influya no sólo el aumento o deficiencia de una clase de inmunoglobulina, sino la relación que existe entre las diferentes subclases.

Considering that some authors have reported an increasing of some immunoglobulins in actinomycetoma patients, in this study we propose to determine differential production of IgG1, IgG2, IgG3, IgG4 and IgGM in 25 patients with actinomycetoma and 25 healthy individuals from a mycetoma endemic area. Immunoglobulins were determined by ELISA technique. To sensibilize the plates, six Nocardia brasiliensis antigens were used: a crude antigen denominated NB and five derivatives (NB2, NB4, NB6, NB8 and NB10) obtained by their isoelectric point. Results showed that all IgG subclasses were higher in the patients’ sera than in control sera, with a maximal difference to IgG3 and IgG4. To the latter subclass, six antigens were highly reactives. IgM levels were similar in both groups. As it occurs in other infections, in the actinomycetoma pathogenesis probably participate the increase or deficiency of a determined immunoglobulin class, as well as the relationship between different subclasses.