A case of a 62-year-old male with Actinomycetoma successfully treated using combined medical and surgical therapy

INTRODUCTION@#Actinomycetoma is a neglected infectious disease that presents with a triad of subcutaneous mass, with sinus formation and seropurulent discharge containing grains. This causes severe functional disability to patients if not treated early and properly.@*CASE REPORT@#We report a case of a 62-year-old adult Filipino farmer diagnosed with actinomycetoma of the left foot. For 8 years, the patient medicated with several antibiotic therapies affording minimal improvement of symptoms. Hence, surgical intervention with combination antimicrobial therapy was done which provided resolution of symptoms, improving the quality of life of the patient.@*CONCLUSION@#Treatment of actinomycetoma must be individualized among patients. Although oral antibiotics became the standard of treatment, combining surgical treatment with oral medications may be considered to ensure effective management of the disease.

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)

Mycetoma caused by Phellinus species: a rare case from Brazil / Micetoma causado por especies de phellinus: un caso raro de Brasil

Abstract Mycetoma is a chronic and slow-developing granulomatous disease characterized by the triad of large painless tumour-like subcutaneous swellings, the formation of sinuses, and discharge that usually contains grains. Phellinus spp. are saprophytic wood-decaying filamentous basidiomycetes. They are an under-recognised cause of invasive fungal infections and are rarely reported worldwide. We report a 59-year-old male patient with mycetoma caused by Phellinus spp. The diagnosis was confirmed with clinical examination, magnetic resonance imaging (MRI) study, soft tissue and bone biopsy culture, and polymerase chain reaction. To the best of our knowledge, this is the first reported case of mycetoma due to Phellinus spp. without chronic granulomatous disease (CGD).

Resumen El micetoma es una enfermedad granulomatosa crónica y de lento desarrollo caracterizada por la tríada de grandes inflamaciones subcutáneas similares a tumores indoloras, la formación de los senos nasales y secreción que generalmente contiene granos. Phellinus spp. son basidiomicetos filamentosos saprofitos que descomponen la madera. Son un poco reconocido causa de infecciones fúngicas invasivas y rara vez se informan en todo el mundo. Presentamos un paciente masculino de 59 años con micetoma causado por Phellinus spp. El diagnostico se confirmó con examen clínico, estudio de resonancia magnética (RM), cultivo de biopsia de tejido blando y óseo y reacción en cadena de la polimerasa. A lo mejor que sepamos, este es el primer caso reportado de micetoma debido a Phellinus spp. sin enfermedad granulomatosa crónica (EGC).

Pseudomycetoma of the scalp caused by Microsporum canis

Abstract Pseudomycetoma is an extremely rare deep mycosis, caused by dermatophytic fungi that penetrate the tissue from infected follicles of tinea capitis. Both clinically and histopathology are similar to eumycetoma, being distinguished through the isolation of the fungus, which in the case of pseudomycetoma can be Microsporum spp. or Trichophyton spp. genre. We present a 24-year-old man with an exuberant tumor in the occipital region with fistula, whose histopathological examination evidenced grains composed of hyaline hyphae and the culture for fungi isolated the agent Microsporum canis. Combined treatment of surgical excision followed by oral griseofulvin for two years was performed, with resolution of the condition.

Diagnóstico de un caso inusual de micetoma por Aspergillus / Diagnosis of an unusual case of Mycetoma due to Aspergillus

Introducción: Las aspergilosis comprenden un amplio y heterogéneo grupo de enfermedades oportunistas causadas por hongos del género Aspergillus, considerados como una causa inusual de infección. Es la causa más frecuente de muerte por neumonía infecciosa e infección diseminada o respiratoria oportunista, en pacientes inmunocomprometidos. Objetivos: Describir las características clínicas de un caso inusual de aspergilosis pulmonar. Caso clínico: Paciente de 56 años de edad con antecedentes personales de hepatopatía alcohólica, ingresado por episodios de expectoración con sangre y tos seca. Se realizaron estudios de laboratorio, imagenológicos y anatomopatológicos que condujeron al diagnóstico de micetoma por Aspergillus fumigatus, lo cual posibilitó indicar el tratamiento adecuado y realizar el seguimiento clínico. Conclusiones: La infección por Aspergillus fumigatus debe ser considerada por el médico de cabecera, debido a que su reporte constituye una herramienta para que pueda establecer una terapéutica temprana y adecuada, dada sus implicaciones pronósticas, su morbilidad y mortalidad en pacientes inmunodeprimidos(AU)

Introduction: Aspergillosis comprises a wide and heterogeneous group of opportunistic diseases caused by fungi of the Aspergillus genus, considered as an unusual cause of infection. It is the most frequent cause of death from infectious pneumonia and disseminated or opportunistic respiratory infection in immunocompromised patients. Objectives: Describe the clinical characteristics of an unusual case of pulmonary aspergillosis. Case report: 56-year-old patient with a personal history of alcoholic liver disease, admitted due to episodes of expectoration with blood and dry cough. Laboratory, imaging, and pathological studies were conducted that led to the diagnosis of mytoma by Aspergillus fumigatus, which made it possible to indicate the appropriate treatment and perform clinical follow-up. Conclusions: Aspergillus fumigatus infection should be considered by the attending physician, because his report constitutes a tool for him to establish an early and adequate therapy, given its prognostic implications and for its morbidity and mortality in immunocompromised patients(AU)

MR Findings of Fungus Ball: Significance of High Signal Intensity on T1-Weighted Images

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.

Eumicetoma e actinomicetoma: uma breve revisão da literatura / Eumycetoma and actinomycetoma: a brief review of the literature

Micetoma é uma infecção que acomete o tecido subcutâneo após a inoculação de microrganismos na pele em locais de pequenos traumas. Caracteriza-se pela ocorrência de tumoração, associada à formação de fistulas e à drenagem de grãos. Trata-se de um grupo de infecções subcutâneas de difícil tratamento com epidemiologia bem definida, acometendo preferencialmente trabalhadores rurais do gênero masculino. Os agentes causadores podem ser fungos ou bactérias. Este artigo propõe-se à revisão dos dados recentes da epidemiologia e tratamento dessas infecções.

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

Abstract: 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.

Mistaken identity: Eumycetoma masquerading as Squamous cell carcinoma

@#We report a case of eumycetoma in a Filipino patient who presented with a solitary reddish brown, moist, multinodular tumor on the dorsum of the left foot of 2 years duration. Biopsy with Periodic acid Schiff (PAS) & Gomori methenamine silver (GMS) staining, fungal culture, ultrasound and X-ray of the foot were done in our institution which confirmed the diagnosis of eumycetoma. The patient was successfully treated with itraconazole 400/day for 3 months, followed by 200mg/day for the succeeding 9 months, leading to complete resolution of the lesion leaving an atrophic hypopigmented scar. A high index of suspicion supported by diagnostic tests aided in the early detection of the disease which also resulted to complete resolution of the disease.

Conjunction of a Fungus Ball and a Pulmonary Tumourlet in a Bronchiectatic Cavity

Herein, we describe the case of a 67-year-old female patient who presented with cough and haemoptysis. Chest computed tomography revealed destruction of the left lower lobe and multiple fungus balls in a bronchiectatic cavity. A left lower lobectomy was performed via thoracotomy. Histopathological examination of the lung showed a concomitant aspergilloma and multiple tumourlets in the large bronchiectatic cavity. Pulmonary intracavitary aspergilloma and concomitant tumourlets are quite rare. Our report presents this interesting case that manifested with haemoptysis.

Pseudomicetoma dermatofítico disseminado em um gato persa - Relato de caso / Dermatophytic pseudomycetoma disseminated in a persian cat - Case report / Pseudomicetoma dermatofitos diseminada en un gato persa - Relato de caso

O pseudomicetoma dermatofítico (PMD) é uma profunda infecção dérmica, causada por fungos, sendo o Microsporum canis o agente etiológico mais frequentemente envolvido. O objetivo do presente trabalho é relatar um caso de pseudomicetoma em um gato persa e alertar os médicos veterinários para incluir a doença como diagnóstico diferencial em dermatopatias em gatos, principalmente os da raça persa. Um felino macho, da raça Persa, de três anos, veio para atendimento apresentando 22 nódulos subcutâneos, distribuídos em todo corpo, com diâmetros que variavam de 2 a 8 centímetros, inclusive dentro do pavilhão auricular direito. Realizou-se raspado cutâneo e citologia aspirativa por agulha fina, obtendo resultados pouco conclusivos. Após a realização de exérese cirúrgica de grande parte dos nódulos e encaminhamento para exame histopatológico, obteve-se o diagnóstico de PMD. Após tratamento com itraconazol 10mg/kg SID por 5 meses houve completa remissão do quadro. A excisão cirúrgica foi essencial para a resolução do quadro, que juntamente com o itraconazol sistêmico tornou o tratamento efetivo para o PMD.(AU)

Dermatophytic pseudomycetoma (DPM) is a deep skin infection. Microsporum canis is the etiologic agent most frequently involved in DPM. The purpose of this study is to report a case of DPM in a Persian cat and suggest the veterinarians to include the disease as a differential diagnosis in skin diseases in Persian cats. A male Persian cat, 3 years old, came to be attended at the Veterinary Hospital. The patient was presenting 22 subcutaneous nodules measuring 2-8 cm in diameter, in different parts of the body, including inside the right ear. Skin scraping and fine needle aspiration cytology were performed, yielding inconclusive results. After performing the surgical removal of most of the nodules and a skin biopsy, the diagnosis of DPM was obtained. The cat had a complete remission of the disease after the treatment with itraconazole 10mg/kg SID for 5 months. It is important to emphasize that, as a therapeutic point of view, surgical excision is considered essential, but not enough to establish a permanent cure. Since there can be recurrence, the systemic use of itraconazole is indicated to efficiently cure animals presenting DPM.(AU)

El pseudomicetoma dermatofitos (PMD) es una infección profunda de la piel causada por hongos, siendo el Microsporum canis el agente etiológico implicado con mayor frecuencia. El objetivo de este trabajo es presentar un caso de pseudomicetoma en un gato persa y alertar los médicos veterinarios para incluir la enfermedad como un diagnóstico diferencial de enfermedades de la piel en gatos, especialmente la raza persa. Un gato macho, de la raza persa, de tres años, llegó a la asistencia presentando 22 nódulos subcutáneos, distribuidos por todo el cuerpo, con diámetros que variaban de 2 a 8 cm, inclusive dentro de la oreja derecha. Se afeitó la piel y con citología por aspiración, con aguja fina, se obtuvo resultados poco conclusivos. Después de realizar la extirpación quirúrgica de la mayor parte de los nódulos y enrutamiento para el examen histopatológico, se ha obtenido el diagnóstico de PMD. Tras el tratamiento con itraconazol l0mg/kg SID, por cinco meses, hubo remisión completa. La escisión quirúrgica fue esencial para la resolución del cuadro, que junto con itraconazol sistémico se convirtió en un tratamiento eficaz para el PMD.(AU)

The use of a documentary drama film to improve the knowledge, attitude and practice of an endemic village population towards mycetoma at Sennar State, Sudan

Background: Mycetoma is a badly neglected tropical disease, characterised by enormous deformities, disfigurement and disabilities if untreated early. Frequently, the majority of the mycetoma patients present late with advanced disease, and the only available treatment for them will be amputation of the affected part. Aim: This study aimed at producing a health promoting film to be used to improve the knowledge, attitude and practice (KAP) of a targeted population in one of the mycetoma endemic villages at Sennar State, Sudan.Materials and Methods : A 26 - minute drama film on a mycetoma patient journey from a small painless mass to advanced disease till lower limb amputation due to missinformation and negligence was performed. Professional actors and cinema work team were employed in the film production. It was filmed in one of the mycetoma endemic areas. A cohort of 250 individuals from two mycetoma endemic villages were included in this study. A closed ended pre-designed questionnaire was used to collect data from the targeted population. The collected data included demographic characteristics, knowledge, attitudes and practices towards mycetoma. The data was collected before and after the film was shown.Results: In this study, 218/250 responded to the questionnare with a response rate of 87%. Fifty-five percent of the respondents were males, 21.6% were farmers, 29.4% were students and 29.4% were unemployed.The film had improved the KAP of the targeted population hence it can be used as a health educational material in other mycetoma endemic areas in the Sudan

Aspergilosis pulmonar en una mujer adulta / Lung aspergillosis in an adult woman

Se describe el caso clínico de una paciente de 44 años de edad, piel mestiza, procedente de zona urbana, con antecedentes personales de hepatopatía alcohólica, quien fue ingresada en el Servicio de Neumología del Hospital General Docente "Dr. Juan Bruno Zayas Alfonso" de Santiago de Cuba, luego de ser remitida de su área de salud por manifestar episodios de expectoración con sangre y tos seca. Se realizaron estudios de laboratorio, cultivo de esputos, rayos X de tórax, tomografía axial computarizada de pulmón y biopsia por aspiración con aguja fina, que condujeron al diagnóstico de micetoma por Aspergillus fumigatus, lo cual posibilitó indicar el tratamiento adecuado y realizar el seguimiento clínico de la afectada.

The case report of a 44 years mixed race patient from an urban area is described, with a personal history of alcoholic liver disease who was admitted in the Pneumology Service of "Dr. Juan Bruno Zayas Alfonso" Teaching General Hospital in Santiago de Cuba, after being referred from her health area due to expectoration episodes with blood and dry cough. Laboratory studies, sputum culture, thorax X rays, lung computerized axial tomography and fine-needle aspiration biopsy were carried out that lead to the mycetoma diagnosis due to Aspergillus fumigatus, which facilitated to indicate the appropriate treatment and to carry out the clinical follow up of the affected patient.

The Imaging Appearance of Sinus Mycetoma: A Case Series / El Aspecto Imagenológico del Micetoma Sinusal: Serie de Casos

Sinus mycetoma is a dense accumulation of hyphae that form a rounded mass within the mucosal confines of a paranasal sinus. The aim of this study was to present a case series of three patients with maxillary sinus mycetoma and to describe their radiographic presentations. Three cases are presented. The first two, a 44-year-old woman and an 88 year-old man, both diabetics, were referred to maxillofacial treatment by other specialties. They both had a bad odor of unknown origin on the facial level and their respective computerized tomographies revealed a unilateral opacification of the maxillary sinus with a region of greater radio-opacity. The third case is a 31-year-old woman with a history of a severe facial trauma who had undergone surgery and for whom orthognathic surgery had been planned to correct side effects. In addition to the orthognathic surgery, the removal of the lesion that appeared opacified in the pre-surgery scan was planned. She showed opacification of the maxillary sinus and during the intraoperative stage, tissue of a whitish-gray appearance was detected on the maxillary sinus. The lesion was completely removed and the result of the histopathological study was sinus mycetoma. The patients evolved favorably and the symptomatology disappeared completely. The growing occurrence of mycetoma and the lack of information on it make publicizing this pathology fundamental so it can be considered as a differential imaging, clinical, and pathological diagnosis and in that way receive adequate and opportune treatment.

El micetoma sinusal es una patología de origen fúngico, que afecta a pacientes inmunocomprometidos, capaz de desorientar en su diagnóstico por su clínica y expresión imagenológica particular. El objetivo de este estudio es presentar una serie de tres casos con micetoma en el seno maxilar y describir su presentación imagenológica. Se presentan tres casos. Los dos primeros, una mujer de 44 años y un hombre de 88 años, ambos diabéticos y derivados a atención maxilofacial por otras especialidades. Coincidía un mal olor expelido de origen desconocido a nivel facial y en sus respectivas tomografías computarizadas se observó velamiento unilateral del seno maxilar con una zona de mayor hiperdensidad. El tercer caso corresponde a una mujer de 31 años con antecedente de trauma facial severo operado, a la cual se le planificó cirugía ortognática para corregir secuelas. En conjunto con la cirugía ortognática, se planificó la biopsia excisional de una lesión hiperdensa que se presentó en el escáner pre-quirúrgico; en el intraoperatorio se pesquisó tejido de aspecto blanquecino grisáceo en el seno maxilar. A todos los pacientes se les realizó la exéresis total de la lesión y el estudio histopatológico dio como resultado micetoma sinusal. Los pacientes evolucionaron favorablemente, desapareciendo por completo la sintomatología. La creciente incidencia del micetoma y el desconocimiento sobre el mismo hacen que sea fundamental la difusión de esta patología para ser considerada como diagnóstico diferencial clínico y patológico, y realizar así un tratamiento adecuado y oportuno.

Stage III eumycetoma successfully treated with oral ketoconazole and surgical debulking

Mycetoma is a chronic, debilitating, granulomatous disease affecting the subcutaneous tissue, fascia, muscle, bone and adjacent organs characterized by triad of tumefaction, draining sinus, and grains. The ten-year incidence in our institution from 2000 to 2010 is 3 out of 81,015 cases. We present a 33-year-old man with a nine-year history of painless nodules and draining sinuses on the left foot which were unresponsive to oral antibiotics and topical antifungals. Biopsy of a nodule was consistent with mycetoma. Fungal culture revealed Madurella mycetomatis growth confirming eumycetoma. X-ray of the left foot showed poorly marginated lucencies on the calcaneus and navicular bones. Ultrasound revealed mixed hyperreflective echoes and multiple small cavities. We diagnosed the patient with Stage III eumycetoma based on the radiographic classification of bone involvement and prescribed oral ketoconazole 400 mg daily for nine months achieving 50-60% decrease in lesion size. Surgical debulking was done and ketoconazole was continued at the same dose for another nine months. There was good granulation tissue formation and no appearance of new lesions after surgery.

A Case of Allergic Fungal Rhinosinusitis with Concurrently Occuring Fungus Ball

Allergic fungal rhinosinusitis (AFRS) is a noninvasive fungal infection of the paranasal sinuses that are usually seen in young immunocompetent patients with atopy and/or asthma. Fungus balls can grow in moist cavities of the paranasal sinuses of a host with normal immunologic status. Cases of AFRS with concurrent fungus balls is very rare. We present a case of a patient who had AFRS on one side of the paranasal sinus and allergic fungal sinusitis on the other side. A 51-year-old female with atopy presented with a few-year history of nasal obstruction and rhinorrhea, as well as a history of high-dose systemic steroid therapy. The patient had nasal polyps and showed an elevated level of total IgE and positive MAST to fungal antigens. Endoscopic sinus surgery was performed. Allergic mucin from the right maxillary sinus contained sheets of eosinophils and Charcot-Leyden crystals. Also, a clay-like dark brown material from the left maxillary sinus was revealed to be a fungus ball.