Fungal mycetoma and pregnancy: An association with costly and difficult management, about a case.

Mycetomas are endemic diseases in tropical and sub-tropical countries of Africa, Asia and America, mainly affecting rural populations living below the poverty line. We report a particular case of a mycetoma associated with pregnancy whose evolution was good, but at the cost of significant financial expenses. This was a 39-year-old woman who developed a fungal mycetoma due to Madurella mycetomatis from the ingunocrural region. The patient had to develop several previous pregnancies on this site of mycetoma, the outcome of which was favorable. The last pregnancy was accompanied by an aggravation of the mycetoma in the form of polyfistulized inflammatory swelling of the right inguino-crural region emitting black grains. Magnetic Resonance Imaging (MRI) of the region showed invasion of the adductor muscles at the level of the root of the thigh on its antero-internal side with no sign of pelvic extension or underlying bone lesion. The patient was treated by surgery associated with antifungal treatment. The evolution was favorable for pregnancy and mycetoma.

A surgeon's trial success of curing pedal eumycetoma.

Mycetoma is a chronic infection of underlying fungal (eumycetoma) or bacterial (actinomycetoma) origin. It is characterised by a clinical triad of tumour-like swelling, actively draining sinuses and macroscopic grains of characteristic colours.We the case of a 66-year-old woman on immunosuppressive therapy presenting with eumycetoma of the foot (Madura foot). The fungal organism cultured was Acrophialophora fusisporaThis case was managed with a combination of extensive surgical debridement, and packing with calcium sulfate (Stimulan) beads impregnated with vancomycin and voriconazole. As far as the authors are aware, this is a novel adjunct to the surgical treatment of deep fungal infection in the foot.Eumycetoma treated with surgery and oral antifungal therapy leads to cure rates of 25%-35%. This novel treatment seems to bear further investigation for the potential to improve cure rates. At 8 months follow-up, our patient appears to be making good progress with no current signs of recurrence.

Mycetoma management and clinical outcomes: the Mycetoma Research Center experience.

BACKGROUND: Mycetoma is a chronic granulomatous inflammatory disease that affects the cutaneous and subcutaneous tissues, leading to gruesome complications if not treated early. As a neglected disease, it has received scant attention in developing curable drugs. Mycetoma treatment is still based on expert opinions in the absence of guidelines. METHODS: This descriptive, cross-sectional, hospital-based study aimed to determine and assess the disease treatment outcomes observed at Mycetoma Research Center, Sudan. RESULTS: In this study, 75% of patients had eumycetoma, all of whom were treated with itraconazole and 37.4% underwent surgical excision, while 25% of the patients had actinomycetoma, 99.2% of whom were treated with a combination of cotrimoxazole and amoxicillin-clavulanate. The cure rate was 12.7% and 14.3% for patients with eumycetoma and actinomycetoma, respectively. Only 6.1% of eumycetoma patients underwent amputation. Remarkably, no patient with actinomycetoma underwent an amputation. Small lesions (OR=10.09, p<0.001) and good follow-up (OR=6.81, p=0.002) were positive predictors of complete cure. In terms of amputation, history of surgical recurrence at presentation (OR=3.67, p=0.020) and presence of grains (OR=7.13, p=0.012) were positive predictors, whereas small lesions were negative predictors (OR=0.06, p=0.009). CONCLUSIONS: Treatment of mycetoma was suboptimal, with a low cure rate despite a long treatment duration. Complete cure has a significant association with small lesions and good follow-up.

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.

Surgery in mycetoma-endemic villages: unique experience.

In this communication, the Mycetoma Research Center (MRC), University of Khartoum, WHO Collaborating Center on Mycetoma, shares its experience in field surgery for mycetoma. The surgery was conducted in two mycetoma-endemic villages in Sennar and the White Nile States in collaboration with local health authorities, local community leaders, activists and civil society associations. In these villages, the local health centres were renovated and operating theatres were established. The medical and health missions' team was established at the MRC. The team conducted 15 missions over the period 2013-2020 and 1200 mycetoma patients received surgical treatment. These included wide local excisions, minor amputations and debridement that were conducted under spinal or ketamine analgesia. The missions adopted a community holistic management approach, which included medical and surgical treatment, health education sessions, village hygiene improvement and socio-economic support. The latter offered the mycetoma amputees artificial prosthesis and financial support. All these services were provided free of charge. This holistic approach proved to be effective for early case detection and management, optimal treatment outcome and favourable disease prognosis. During the study period, the number of patients with massive lesions and the amputation rate decreased and this reduced the medical and socio-economic disease burdens on patients and families. This treatment approach needs the collaboration of all stakeholders for sustainability and quality improvement.

Eumycetoma, A Neglected Tropical Disease in the United States.

Eumycetoma, caused by fungi, is a neglected tropical disease. It is endemic in the "mycetoma belt" countries but rare in North America. We report a case of pedal eumycetoma in the state of Maryland. A 51-year-old male immigrant from Guatemala presented with multiple, enlarging nodules on the dorsal surface of his left great toe present for 1 year, and a new one in the left arch area present for 6 months. The nodular lesions were surgically excised in two separate operations. Pathologic evaluation of all nodules revealed eumycetomas characterized by the Splendore-Hoeppli phenomenon, showing an amorphous eosinophilic center filled with numerous fungal hyphae, observed on periodic acid-Schiff-stained slides, with a surrounding cuff of neutrophils. Polymerase chain reaction-based sequencing identified Cladosporium cladosporioides in the tissues. The patient was further treated with oral fluconazole for 2 months. The patient recovered well postoperatively and had no recurrence at 20-month follow-up. In conclusion, even though eumycetoma is regarded as a rare disease in North America, its incidence may be higher than reported because of millions of immigrants from endemic regions in the United States, which highlights the need to raise awareness of this devastating disease in the medical community. Eumycetoma needs to be differentiated from other infectious and noninfectious benign and malignant lesions. Optimal treatment includes surgical excision with antifungal therapy.

Madura's foot: reasons for the delay in diagnosis and consequences for the management (a case report).

Madura foot, relatively easy to diagnose in tropical countries, is very rare and unrecognized in Morocco, causing diagnostic delays. We present the case of a 54-year-old patient with mycetoma for 3 years who initially consulted two general practitioners, then an endocrinologist and finally a dermatologist in order to be diagnosed correctly. The diagnosis of mycetoma based on biological criteria was established at a late stage of irreversible bone lesions; requiring amputation by the orthopedic team. Mycetomas are fungal or bacterial. Delays in diagnosis and care are frequent in Morocco. The diagnosis is based on biology; however, radiological examinations are necessary to assess the extension. The initial treatment is medicinal. Surgery takes place in late stages.

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.

Unusual extra-podal fungal mycetoma with black grains in a Senegalese child.

Mycetoma remains endemic in the tropical and subtropical regions of the "mycetoma belt" including Senegal. It affects more commonly young men in the age group of 20 to 40 years. The foot represents the most commonly affected site. The most common extra-podal localizations are leg, knee, buttocks, hand and arm. We report an exceptional case of cervical fungal mycetoma that occurred in a 13-year-old Senegalese child. He consulted for a cervico-submandibular tumefaction with multiple sinuses and black grains discharge evolving since 6 years, associated to laryngeal dyspnoea. Mycological examination with culture isolated Madurella mycetomatis. Cervical CT Scan showed bone and soft tissue invasion. Terbinafine alone was administered. During the evolution, tracheotomy was performed following the aggravation of the laryngeal disorders. Death from severe sepsis occurred after 8 months of evolution. The particularities of our case are the occurrence of fungal mycetoma in a child, the cervical localization and the difficulties of therapeutic management largely due to the diagnostic delay.

Surgical Management of Mycetoma in Chad: Experience from the French Forward Surgical Team Deployed in N'Djamena.

Mycetoma is a disease that occurs in the mycetoma belt, between latitudes 15̊ south and 30̊ north. It affects disadvantaged regions with limited access to medical and health facilities. Its general principles of care have changed little and are poorly known. We analyzed the management of mycetoma in Chad by French military surgeons deployed within the Epervier and Barkhane operations. This retrospective descriptive study was conducted among the cohort of Chadian patients managed by the N'Djamena forward surgical team from 2007 to 2018 as part of the medical support to the population. It includes 132 patients who had surgery for mycetoma. Surgical parameters of primary treatment and revisions procedures were analyzed. Postoperative follow-up was at least six months. Amputation was performed in 87/132 (66%) patients. Overall 11 (8.3%) required revision surgery, including 7 (5%) with eumycetoma recurrence. All recurrences occurred in the lower limb. The recurrence rate after excision was 10.2% (5/49) versus 2.3% after amputation (2/87). In the absence of effective and accessible medical treatment, surgery remains the basic treatment for mycetoma. Salvage surgery with local excision should always be considered. However, amputation is the only reliable treatment in cases with late presentation. It should not be proposed too early as limb function is preserved for a long time.

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.

Madurella mycetomatis infection of the buttock in an Eritrean refugee in Switzerland: a case report.

BACKGROUND: Mycetoma is a neglected infectious disease caused by a fungus (eumycetoma) or bacteria (actinomycetoma); it is characterized by chronic local inflammation with sinus formation and purulent discharge. Its course can be quite devastating because of the difficulty in diagnosing the infection and in eliminating the causative agent. Although endemic in many countries in the tropics and subtropics, the migration of Africans to Europe may increase the presence of this neglected disease in European countries. We present a case of an Eritrean patient living in a non-endemic country who was diagnosed as having an infection of Madurella mycetomatis in an atypical location in his body. CASE PRESENTATION: We report the case of a 35-year-old African male refugee from Eritrea, living in Switzerland since 2015, who presented with a 1-year history of a painful soft tissue swelling associated with dark nodules in his right buttock. He mentioned having several previous surgeries after 2001 while he was in Eritrea due to recurrent abscess formation on this body area. In the previous months, the swelling had become more significant and nodules started draining a purulent fluid. An initial diagnostic hypothesis of buttock abscess was made and he was referred to a dermatologist for diagnostic confirmation and further specialist care due to the size and atypical presentation. After a punch biopsy, the diagnosis of eumycetoma was confirmed and cultures developed Madurella mycetomatis. The initial treatment approach consisted of oral treatment by itraconazole; however, a surgical resection of the lesions was finally needed. CONCLUSIONS: Although rare, mycetoma should be diagnosed as early as possible to avoid long-lasting complications. Primary care physicians in European countries are frequently in the first line of care of migrant patients and therefore should be aware of the common and uncommon clinical presentations of mycetoma.

[Recidives of Mycetoma after Amputation in Dakar (Senegal)]. / Récidives de mycétome après amputation à Dakar (Sénégal).

The treatment of fungal mycetoma is essentially surgical. This carcinological-like surgery consists of amputation in case of bone involvement. The recurrences after amputation are rare and address the problem of the operative indication. We report 5 cases of recurrence of fungal black-grain mycetoma after amputation of leg or thigh. Case 1: a 52-year-old patient with a mycetoma of the knee evolving for 8 years. There is no history of surgery. A thigh amputation with ganglion dissection is performed. One year after the surgical procedure, the patient presents a recurrence on the amputation stump and on the lymph node dissection site. An indication of hip disarticulation is made and performed 17 months after amputation. Case 2: a 25-year-old patient who has a black-grain mycetoma of the foot with osteitis evolving since 10 years. A leg amputation was performed. The patient had a recurrence at the popliteal level at 15 months postoperatively. An indication of amputation of the thigh is posed and refused by the patient. Case 3: a30-year-old woman with black-grain mycetoma of the knee with bone involvement for more than 10 years. A thigh amputation was performed and at nine months postoperativeshe presented a recurrence in the amputation stump. She was lost of sight despite the decision of surgical revision. Case 4: a 43-year-old patient operated on his foot and leg mycetoma at least 5 timesbefore amputation in 2000. The recurrence occurred one year after amputation. 18 years after amputation, a new surgical procedure was difficult due to extension of the lesions in the pelvis. Case 5: a 50-year-old female patient operated in Mauritania in 2012 (thigh amputation for mycetoma of the knee). She presented a recurrence on the amputation stump in 2018. An indication of disarticulation of the hip was posed and refused by the patient. These recurrences were testified by to the persistence of grains on the preserved segment. They pose the problem of the level of amputation and therefore of preoperative planning. Good preoperative planning allows optimization of the surgical procedure and avoids certain recurrences.

La chirurgie constitue le temps essentiel du traitement des mycétomes fongiques. Elle consiste en une amputation en cas d'atteinte osseuse. Nous avons observé 5 cas de récidives après amputation pour mycétome. Il s'agit dans tous les cas de patients présentant des mycétomes à grain noir avec atteintes osseuses. Les récidives sont survenues à moins de 18 mois de l'amputation faisant parler de reprise évolutive et posant le problème du niveau de l'amputation.

Pedal Fungal Mass in the Midwest.

Mycetoma cases are predominantly found in tropical regions and are a rare finding in the United States. These masses that are fungal or bacterial in origin can result in significant destruction of soft tissue and bone. We present a case of a patient who emigrated from Mexico to Indianapolis. He presented with a soft-tissue mass that was excised and ultimately found to be a eumycetoma of the hallux of his left foot. Successful treatment included surgical resection in combination with postoperative terbinafine, which was pulse dosed to decrease its impact on hepatic function.