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

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

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.

Abdominal wall actinomycotic mycetoma involving the bowel: a rare entity.

Actinomycotic mycetoma is a disease of the tropical region and usually presents as a chronic, suppurative and deforming granulomatous infection. We present an unusual case of actinomycotic mycetoma of the abdominal wall that was found to infiltrate into the bowel. A 51 year-old man presented with pain and swelling in the left flank of 2-year duration. Even after comprehensive preoperative evaluation with advanced radiological imaging, biochemistry and pathology, the diagnosis could not be arrived at. Histopathological examination of the excised specimen after the surgery guided to the diagnosis of actinomycotic mycetoma, which entirely changed the management in the postoperative period. We propose that mycetoma should be kept as a possible differential diagnosis for anterior abdominal wall swelling in the indicated clinical setting and the investigations be done keeping the same in mind. Otherwise, a lot of valuable time may be lost allowing the disease to progress further.

A Rare Case of Fungal Rhinosinusitis Caused by Scedosporium apiopermum.

Fungal rhinosinusitis (FRS) is inflammation of the paranasal sinus mucosa due to fungal infections, which can be invasive or non-invasive. The occurrence of a sphenoid mucocele with a fungal ball is rare. We report a case of sphenoid sinus mucocele with a fungal ball caused by Scedosporium apiopermum in a 32-year-old female who presented to the Emergency Department with persistent headache not relieved on medications. The radiological images showed a mucocele with clival osteomyelitis. Urgent endoscopic examination and debridement was undertaken which demonstrated a mucocele with fungal ball. Microbiological examination confirmed it to be Scedosporium apiopermum.

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.

Pseudomycetoma of the scalp caused by Microsporum canis.

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.

Proteomic analysis of the processes leading to Madurella mycetomatis grain formation in Galleria mellonella larvae.

Mycetoma is a neglected chronic and granulomatous infection primarily associated with the fungal pathogen Madurella mycetomatis. Characteristic of this infection is the formation of grains. However, the processes leading to grain formation are not known. In this study, we employed a proteomic approach to characterise M. mycetomatis grain formation in Galleria mellonella larvae and map the processes leading to grain formation over time. For this, at 1 day, 3 days and 7 days post-inoculation, proteins from grains and hemolymph were extracted and analysed by label-free mass spectrometry. A total of 87, 51 and 48 M. mycetomatis proteins and 713, 997, 18 G. mellonella proteins were found in grains on day 1, 3 and 7 post-inoculation respectively. M. mycetomatis proteins were mainly involved in cellular metabolic processes and numerous enzymes were encountered. G. mellonella proteins were primarily involved in the nodulation process. The proteins identified were linked to nodulation and grain formation and four steps of grain formation were identified. The results of this proteomic approach could in the future be used to design novel strategies to interfere with mycetoma grain formation and to combat this difficult to treat infection.

Acremonium nail bed mycetoma masquerading as subungual squamous cell carcinoma.

Acremonium is a large fungal genus that is comprised of approximately 150 species, found ubiquitously in nature. Although the majority are recognized as being saprophytes in soil and pathogens of plants, several species are emerging as causative agents of a variety of human infections, including mycetomas. Herein, we present a young man that was referred to our department with a painful subungual mass that developed following traumatic inoculation of Acremonium spp. In recent years, the role of Acremonium spp. has been increasingly recognized in localized infections, such as mycetoma, in humans. Other locally invasive as well as disseminated infections are also described. Optimal treatment of Acremonium spp. mycetoma is not well defined owing to the rarity of cases, thus posing a therapeutic challenge.

Clinical characteristics and treatment of actinomycetoma in northeast Mexico: A case series.

BACKGROUND: Mycetoma is a neglected tropical disease characterized by nodules, scars, abscesses, and fistulae that drain serous or purulent material containing the etiological agent. Mycetoma may be caused by true fungi (eumycetoma) or filamentous aerobic bacteria (actinomycetoma). Mycetoma is more frequent in the so-called mycetoma belt (latitude 15° south and 30° north around the Tropic of Cancer), especially in Sudan, Nigeria, Somalia, India, Mexico, and Venezuela. The introduction of new antibiotics with fewer side effects, broader susceptibility profiles, and different administration routes has made information on actinomycetoma treatment and outcomes necessary. The objective of this report was to provide an update on clinical, therapeutic, and outcome data for patients with actinomycetoma attending a reference center in northeast Mexico. METHODOLOGY/PRINCIPAL FINDINGS: This was a retrospective, cross-sectional, descriptive study of 31 patients (male to female ratio 3.4:1) diagnosed with actinomycetoma by direct grain examination, histopathology, culture, or serology from January 2009 to September 2018. Most lesions were caused by Nocardia brasiliensis (83.9%) followed by Actinomadura madurae (12.9%) and Actinomadura pelletieri (3.2%). About 50% of patients had bone involvement, and the right leg was the most commonly affected region in 38.7% of cases. Farmers/agriculture workers were most commonly affected, representing 41.9% of patients. The most commonly used treatment regimen was the Welsh regimen (35.5% of cases), a combination of trimethoprim/sulfamethoxazole (TMP/SMX) plus amikacin, which had a 90% cure rate, followed by TMP/SMX plus amoxicillin/clavulanic acid in 19.4% of cases with a cure rate of 100%. In our setting, 28 (90.3%) patients were completely cured and three (9.7%) were lost to follow-up. Four patients required multiple antibiotic regimens due to recurrences and adverse effects. CONCLUSIONS/SIGNIFICANCE: In our sample, actinomycetoma was predominantly caused by N. brasiliensis. Most cases responded well to therapy with a combination of TMP/SMX with amikacin or TMP/SMX and amoxicillin/clavulanic acid. Four patients required multiple antibiotics and intrahospital care.

Madurella mycetomatis infection of the foot: a case report of a neglected tropical disease in a non-endemic region.

BACKGROUND: Mycetoma is an uncommon chronic granulomatous infection of cutaneous and subcutaneous tissues that can be caused by filamentous bacteria (actinomycetoma) or fungi (eumycetoma). It is the prerogative of young men between the third and fourth decade and is transmitted through any trauma causing an inoculating point. The classic clinical triad associates a painless hard and swelling subcutaneous mass, multiple fistulas, and the pathognomonic discharge of grains. Although endemic in many tropical and subtropical countries, mycetoma can also be found in non-endemic areas as in Morocco, and causes then diagnosis problems leading to long lasting complications. Therefore, we should raise awareness of this neglected disease for an earlier management. Under medical treatment however, mycetoma has a slow healing and surgery is often needed, and relapses are possible. CASE PRESENTATION: Herein we report a case of a 64 years old patient, with a history of eumycetoma occurring ten years ago treated with oral terbinafine coupled with surgery. A complete remission was seen after 2 years. He presented a relapse on the previous scar 6 months ago. There wasn't any bone involvement in the magnetic resonance imaging (MRI). The patient was put under oral terbinafine with a slow but positive outcome. CONCLUSION: Through this case report, we perform a literature review and highlight the importance of increase awareness of mycetoma in clinical practice especially in non-endemic regions.

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

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.

The Accuracy of Histopathological and Cytopathological Techniques in the Identification of the Mycetoma Causative Agents.

Mycetoma is a devastating neglected tropical disease, caused by various fungal and bacterial pathogens. Correct diagnosis to the species level is mandatory for proper treatment. In endemic areas, various diagnostic tests and techniques are in use to achieve that, and that includes grain culture, surgical biopsy histopathological examination, fine needle aspiration cytological (FNAC) examination and in certain centres molecular diagnosis such as PCR. In this retrospective study, the sensitivity, specificity and diagnostic accuracy of grain culture, surgical biopsy histopathological examination and FNAC to identify the mycetoma causative organisms were determined. The histopathological examination appeared to have better sensitivity and specificity. The histological examination results were correct in 714 (97.5%) out of 750 patients infected with Madurella mycetomatis, in 133 (93.6%) out of 142 patients infected with Streptomyces somaliensis, in 53 (74.6%) out of 71 patients infected with Actinomadura madurae and in 12 (75%) out of 16 patients infected with Actinomadura pelletierii. FNAC results were correct in 604 (80.5%) out of 750 patients with Madurella mycetomatis eumycetoma, in 50 (37.5%) out of 133 Streptomyces somaliensis patients, 43 (60.5%) out of 71 Actinomadura madurae patients and 11 (68.7%) out of 16 Actinomadura pelletierii. The mean time required to obtain the FNAC result was one day, and for the histopathological examinations results it was 3.5 days, and for grain it was a mean of 16 days. In conclusion, histopathological examination and FNAC are more practical techniques for rapid species identification than grain culture in many endemic regions.

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.

Interleukin-17 and matrix metalloprotease-9 expression in the mycetoma granuloma.

Mycetoma is a persistent, progressive granulomatous inflammatory disease caused either by fungi or by bacteria. Characteristic of this disease is that the causative agents organise themselves in macroscopic structures called grains. These grains are surrounded by a massive inflammatory reaction. The processes leading to this host tissue reaction and the immunophenotypic characteristics of the mycetoma granuloma are not known. Due to the massive immune reaction and the tissue remodeling involved, we hypothesised that the expression levels of interleukin-17 (IL-17) and matrix metalloprotease-9 (MMP-9) in the mycetoma granuloma formation were correlated to the severity of the disease and that this correlation was independent of the causative agent responsible for the granuloma reaction. To determine the expression of IL-17 and MMP-9 in mycetoma lesions, the present study was conducted at the Mycetoma Research Centre, Sudan. Surgical biopsies from 100 patients with confirmed mycetoma were obtained, and IL-17 and MMP-9 expression in the mycetoma granuloma were evaluated immunohistochemically. IL-17 was mainly expressed in Zones I and II, and far less in Zone III. MMP-9 was detected mainly in Zones II and III, and the least expression was in Zone I. MMP-9 was more highly expressed in Actinomadura pelletierii and Streptomyces somaliensis biopsies compared to Madurella mycetomatis biopsies. MMP-9 levels were directly proportional to the levels of IL-17 (p = 0.001). The only significant association between MMP9 and the patients' characteristics was the disease duration (p<0.001). There was an insignificant correlation between the IL-17 levels and the patients' demographic characteristics.

Mycetoma in China: A Case Report and Review of the Literature.

Mycetoma is a chronic granulomatous infectious disease that can affect the skin, subcutaneous tissue, fascia and bone. It can be caused by filamentous bacteria or fungi and usually involves the legs and feet. Mycetoma is endemic in tropical and subtropical regions and is easily misdiagnosed in clinical practice because of its nonspecific clinical features and lack of awareness of the disease. Although mycetoma is very rare in mainland China, an increasing number of cases have been reported in recent years. Here, we report a case of mycetoma in a patient who was misdiagnosed many years before receiving the correct treatment, leading to disease progression and motion limitation. The grains that represent microorganismal colonies were important clues for diagnosis. We also reviewed reported cases of mycetoma in mainland China. The majority of cases were reported from southern regions. Actinomycetoma was more commonly reported than was eumycetoma. The causative agents of actinomycetoma included Nocardia brasiliensis, N. asteroides, N. otitidiscaviarum, N. ninae and Gordonia terrae, and the causative fungi of eumycetoma were identified as Madurella mycetomatis, Fonsecaea pedrosoi and Acremonium falciforme. Notably, the diagnosis of mycetoma was delayed from months to decades in all of the patients, likely due to a lack of clinical experience. Our literature review suggests the importance of increased awareness of mycetoma in clinical practice, especially in non-endemic regions. Further investigative studies are needed to determine the real incidence of the disease in China.

Mycetoma: reviewing a neglected disease.

Mycetoma caused by either filamentous fungi (eumycotic) or bacteria (actinomycotic) has recently been recognized by the World Health Organization as a neglected tropical disease. Although mycetoma is preventable and treatable, especially in the early stages, it carries high morbidity and a huge socioeconomic burden. Skin and subcutaneous tissue is affected, with a classic presentation of hard woody swellings, discharging sinuses and presence of grains (containing the causative organism). Variants with swelling without sinuses have also been described. Left untreated it may involve underlying bone and muscle, leading to permanent disability. Common actinomycotic species include Streptomyces somaliensis, Actinomadura madurae, Actinomadura pelletieri, Nocardia brasiliensis and Nocardia asteroides, while Madurella mycetomatis, Madurella grisea, Pseudoallescheria boydii and Leptosphaeria senegalensis are common eumycotic agents. Men are more commonly affected than women, and the leg is the most frequently affected site. Diagnosis in suspected lesions is made with the help of grain examination, microscopy, imaging (radiography, ultrasonography, magnetic resonance imaging) and culture, and more recently by molecular methods such as PCR and molecular sequencing. Molecular sequencing for both fungi and bacteria is important for rapid and correct diagnosis, especially in culture-negative cases. Treatment is long, more successful in actinomycetoma than eumycetoma, and may require a holistic approach comprising antimicrobials, surgery and rehabilitation. Mycetoma can be prevented by simple measures such as wearing protective garments and shoes, especially in rural areas and during outdoor activities.

Immigration Brings New Pathology with No Standardized Treatment Protocol Madura Foot Case Studies Phialemonium and Phaeoacremonium.

Madura foot is an uncommon invasive soft-tissue infection that foot and ankle specialists encounter. We present two rare cases of Phialemonium and Phaeoacremonium fungi infections of the foot diagnosed in northern California to inform physicians on the presentation and current treatment options for this unique pathology. The two cases presented outline the clinical presentations, diagnostic data, and surgical and antimicrobial interventions. There is a concentration on the antimicrobial options depending on which of the over 20 species is encountered. The pertinent literature and supporting data are reviewed to create an outline for discussion of treatment protocols when faced with these emerging opportunistic infections.

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.