RESUMEN
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.
Asunto(s)
Granuloma Laríngeo/diagnóstico , Madurella , Micetoma/diagnóstico , Adolescente , Niño , Diagnóstico Tardío , Disnea/diagnóstico , Disnea/microbiología , Disnea/cirugía , Resultado Fatal , Granuloma Laríngeo/tratamiento farmacológico , Granuloma Laríngeo/microbiología , Granuloma Laríngeo/cirugía , Humanos , Madurella/crecimiento & desarrollo , Madurella/aislamiento & purificación , Micetoma/tratamiento farmacológico , Micetoma/microbiología , Micetoma/cirugía , Senegal , Sepsis/diagnóstico , Sepsis/microbiología , Terbinafina/uso terapéutico , Cartílago Tiroides/microbiología , Cartílago Tiroides/patología , Cartílago Tiroides/cirugía , TraqueotomíaRESUMEN
UNLABELLED: We report the case of a fungal mycetoma due to Madurella mycetomatis that failed to respond to surgery and antifungal treatment but responded strongly to the addition of a non-steroidal anti-inflammatory drug (NSAID). This African patient was born in Mauritania in 1972. He was a herdsman, living close to the Senegal River. The first nodules appeared on the left foot at the age of 13years (1985). The patient suffered frequent flare-ups with the appearance of black grains and underwent surgery in 1988 and 1992 in Senegal. After remission for several months after surgery, new fistulae occurred. The patient emigrated to France in 1995 and underwent a third surgical intervention in 1996. M. mycetomatis was cultured from the black grains. The patient was otherwise in good health, with no diabetes, and HIV tests were negative. We saw the patient for the first time in 2005, at which time he had flare-ups every two to three months. Imaging disclosed an absence of bone involvement. The patient underwent a fourth operation in October, 2005, and voriconazole treatment was initiated. A new flare-up occurred in February, 2006. CT, MRI, and PET scans revealed calcaneus and tarsal involvement, and posaconazole then replaced voriconazole. Flucytosine was added four months later, due to an absence of improvement. New flares-ups occurred and a fifth surgical intervention was performed in September, 2006. The pain, which had been present for three years, worsened; the patient had to stop working and was no longer able to walk without crutches. Amputation of the foot was considered. Empiric treatment with a NSAID, diclofenac (Voltaren(®); 100mg/day), was added to the antifungal treatment in November 2006, to treat the patient's pain and inflammation. A major improvement was observed within one week. The patient was able to walk without crutches one month later. After two months, clinical examination was normal: no pain, inflammation, nodules or fistulae. Flucytosine was stopped after six months of treatment, in January 2007, diclofenac after 10months, in October 2007, and posaconazole after 18.5months, also in October 2007. No relapse has occurred during the eight years of follow-up since treatment ended. The patient seems to have been cured and has normal CT, MRI, and PET scans. IN SUMMARY: This eumycetoma, which had progressed over 20years despite surgery and antifungal treatments, seems to have been cured by the addition of a NSAID. This observation suggests that inflammation plays a major role in the pathogenesis of fungal mycetoma. Clinical studies of treatments including an NSAID should be conducted to confirm this finding.
Asunto(s)
Antiinflamatorios no Esteroideos/uso terapéutico , Madurella , Micetoma/tratamiento farmacológico , Adolescente , Antifúngicos/uso terapéutico , Humanos , Madurella/aislamiento & purificación , Madurella/patogenicidad , Masculino , Mauritania , Micetoma/diagnóstico , Micetoma/microbiología , Micetoma/patología , Inducción de Remisión , Senegal , Insuficiencia del TratamientoRESUMEN
Black-grain mycetomas are fungal and cannot be healed by antifungals only.We conducted this study to determine the interest of carcinologic surgery only in the management of those black-grain mycetomas.We led a retrospective study concerning 44 patients over a period of 52 months. The mean age of patients was 32.5 years. The main location of mycetomas was the foot (21 cases), the ankle (10 cases), the knee (8 cases), the leg (3 cases), the chest, the back and the pulp of the fourth finger once each. The bone was affected in 21 cases. The surgery consisted in a carcinologic excision or amputation depending on the extension of the lesion and the degree of bony infringement, with ganglionic cleaning out in case of lymphadenopathy. In the average retrospect of 25.5 months, we noted 8 cases of recurrences among which 2 on the amputated patients and 6 on the patients treated by carcinologic excision. The surgery constitutes the main therapeutic issue of the fungal mycetomas.
Asunto(s)
Micetoma/cirugía , Adolescente , Adulto , Anciano , Femenino , Hospitales , Humanos , Masculino , Persona de Mediana Edad , Micetoma/epidemiología , Estudios Retrospectivos , Senegal/epidemiología , Adulto JovenRESUMEN
Fungal mycetoma are inflammatory pseudo-tumors of subcutaneous tissues and possibly bones due to exogenous fungi. They have a chronic course, often poly-fistulated with an emission of fungal grains. We report the case of a 65-year-old farmer with a thoracic fungal mycetoma discovered incidentally, associated with bone involvement. The diagnosis was confirmed by the positive culture to Madurella mycetomatis. The outcome was favorable with terbinafine 1g per day for 12 months associated with complete excision of oncologic type followed by a skin graft.