Mycetoma: retrospective study of 13 cases in Tunisia

Mycetomas are inflammatory pseudotumors of chronic and progressive evolution, of fungal (Eumycetoma), or bacterial (Actinomycetoma) origin. We conducted a retrospective study of patients who have been consulted for mycetoma over a period of 28 years. Thirteen cases of mycetomas were collected during the 28-year period, 10 patients were of rural origin. The lesions were localized on the foot in all instances. There was no male predominance. Histopathological examination confirmed mycetoma in all 13 cases and the determination of the infectious agent in 9 cases: 4 actinomycotic and 5 fungal. Microbiological examination identified the species in 7 cases. Bone involvement was found in 10 cases. Eumycetomas were treated by ketoconazole. The other cases were treated as actinomycetomas by antibiotics. The antibiotic treatment was associated with surgical excision in 4 cases and with amputation in 1 case. Mycetomas are rare in Tunisia, and only observed sporadically, for this reason the diagnosis is usually late, with severe functional, psychological, and socioeconomic consequences.     

Restoration of bones in mycetoma.

In a case of actinomycotic mycetoma caused by Streptomyces madurae with extensive destruction of the bone, the foot was saved from amputation by appropriate antibiotic therapy given for a period of 2 1/2 years. Periodic x-ray examinations gave a valuable guide to the progress of the patient. Chemotherapy and antibiotics play an important role in the present day management of mycetoma. Cutaneous amelioration is not the criteria for cure. The bone restoration that occurs much later should be the guide for the duration of therapy.     

Does the Benefit of Salvage Amputation Always Outweigh Disability in Drug-Failure Mycetoma?: A Tale of Two Cases

It is popularly believed that eumycetoma cases should be dealt with using surgical amputation for a better chance of cure especially when chemotherapy has failed. However, amputation leads to disability on one hand and on the other it may also fail to be curative. We present two cases with contrasting treatment options and outcome. In the eumycetoma case reported here, a 40-year-old male presented with right foot swelling for 16 years, from which Scedosporium apiospermum was isolated. He responded poorly to antifungal therapy and refused below-knee amputation 12 years ago. With counseling and wound care his condition improved, and Foot and Ankle Ability Measure (FAAM) score remained almost stable at 90% for 16 years, which is much better than the average functional outcome after amputation. Another 46-year-old female underwent below-knee amputation after receiving incomplete courses of antibiotics and antifungals for mycetoma of unknown etiology. She presented to us after recurrence of mycetoma on an amputated stump and was successfully treated by proper courses of antibiotics after detecting the causal agent, Actinomadura madurae. Her post-amputation disability and depression could have been avoided if the hasty decision of amputation had not been taken. In our opinion, living with drug-non-responsive mycetoma, supported by symptomatic management, may be a better option than amputation and its associated morbidities. So before taking the path of salvage amputation, we must consider many aspects, including patient''s livelihood, psychological aspects and chances of recurrence even after the procedure.     

Tumoral mycetoma of the buttock

BACKGROUND: Mycetomas are actinomycosic or fungal infections where the infectious agent produces grains. We report an atypical case of fungal mycetoma presenting as a tumoral formation on the buttock. CASE REPORT: A 56-year-old unemployed man from the Diourbel region of central Senegal consulted in February 1997 for a fistulalized tumor of the right buttock which had developed spontaneously and progressed for 5 years. The patient's general health remained satisfactory. Physical examination showed a voluminous 25 cm tumefaction extending from the right buttock to the perineum. The tumor showed a few areas of fistualization which discharged black grains under pressure. There was a 1.5 cm right inguinal node which did not appear to be inflammatory. The remainder of the physical examination was normal. Pathology reported inflammatory connective tissue surrounding a brownish polycyclic grain composed of spores and mycele filaments. The diagnosis of fungal mycetoma was retained and surgical excision under general anesthesia was performed. DISCUSSION: This is an atypical case of fungal mycetoma because of its tumoral form and gluteal localization. The differential diagnosis was cutaneous neurofibroma, myoma, or Darier-Ferrand dermatofibrosarcoma. The frequency of extrapodal red grain mycetomas has been pointed out by several authors from Senegal, but extrapodal black grain forms as seen in our case are more exceptional.     

Epidemiological and Etiological Study of Foot Mycosis in Tunisia

BackgroundTinea pedis and onychomycosis are among the commonest fungal diseases in the world. Recently, there has been an increase in the numbers of fungal agents implicated in these conditions.ObjectiveTo analyze the epidemiology of fungal foot diseases and to identify associated etiological factors in outpatients attending the Department of Dermatology of Charles Nicolle Hospital in Tunis, Tunisia.Patients and MethodsOne hundred and forty eight patients were assessed for the presence of fungal foot diseases during the period between January and April 2009. The mean age was 41.5 years (range: 2–87 years) and sex ratio was 0.8. A complete dermatological examination was performed on all subjects, and specimens of the feet were taken from patients presenting signs of tinea pedis or onychomycosis for microscopy and fungal culture.ResultsFungal foot infection was suspected in 71 subjects, and the diagnosis was confirmed in 67 cases (45.3%) by positive microscopy or culture. Older age and family history of mycosis were predisposing factors for foot fungal infection. The condition was caused by dermatophytes in 57.1% of cases and Candida species in 35.7%. Trichophyton rubrum and Candida parapsilosis were the predominant dermatophyte and yeast species, respectively.     

Mycetoma caused by Petriellidium boydii: treatment with ketoconazole

A case of mycetoma of the lower leg (Madura foot) with bone involvement caused by Petriellidium boydii is presented. After an initially favourable therapeutic response to ketoconazole (2 X 200 mg/die) the process recurred despite proven sensitivity of the fungus in vitro. It could not be controlled with conservative treatment and ultimately necessitated amputation of the lower leg. A striking, hitherto unreported feature of mycetoma was the episodic occurrence of circulating immune complexes with febrile monoarthritis of the adjacent ankle.     

Pseudallescheria boydii Mycetoma in Northern New England

Mycetoma is a chronic subcutaneous fungal infection characterized by tumefaction, draining sinuses, and grains. It is most common in the tropics but occasionally occurs in the United States. We report a case of a mycetoma affecting the foot of a 38-year-old mentally retarded man from northern New England. The causal organism was identified as Pseudallescheria boydii, the most common cause of mycetoma in the United States. The patient showed a partial response to 8 months of ketoconazole therapy.     

Mycetoma (madura foot) in the United Kingdom–a survey of forty-four cases

Forty-four cases of mycetoma (madura foot) presenting in the United Kingdom between 1963 and 1981 have been analysed. The majority of the patients originated from the tropics particularly the West Indies (41%). The commonest organisms isolated belonged to the pale grain eumycetoma (true fungal mycetoma) group, particularly Petriellidium boydii or Fusarium spp. However, in four instances the infecting organism was not identified because of a lack of distinguishing characteristics. All seven patients from the Middle East had actinomycetoma infections caused by Streptomyces somaliensis. The diagnosis and management of three patients are discussed in more detail.     

Perianal actinomycetoma experience of 20 cases

BACKGROUND: Actinomycetoma is a chronic infection resulting from aerobic Actinomycetes. The major agents are Nocardia brasiliensis, Actinomadura madurae, and Streptomyces somaliensis. The most frequent topographies are the lower and upper limbs. The prognosis of this disease is determined by several factors, such as etiologic agent, clinical topography, and depth of disease (degree of involvement, visceral, and bone affection). The purpose of this paper was to present our experience with actinomycetoma of the perianal region. METHODS: This study comprises 20 cases of perianal actinomycetoma, all of which were clinically and microbiologically proven by direct examinations, cultures, and biopsies. Clinical responses to the two principal treatment regimes used [combination of trimethoprim-sulfamethoxazole (TMS/SMX) and diaminodiphenylsulfone (DDS) or amikacine plus TMS/SMX] are reported. RESULTS: Most of the cases were male (17/20, 85%), the mean age was 42.1 years, and the farmers predominated (90%). The principal etiologic agent isolated was N. brasiliensis (85%). CONCLUSIONS: Perianal actinomycetoma is a rare entity. Differential diagnosis with anal sinuses, hydroadenitis, and cutaneous tuberculosis must be made in endemic areas by performing mycologic tests and biopsies. Treatment depends on the etiologic agent involved and the patient's condition.     

Chromoblastomycosis

Chromoblastomycosis, together with phaeohyphomycosis and mycetoma, makes up the disease entities caused by the dematiaceous fungi. Most cases of chromoblastomycosis are caused by five genera of fungi: Fonsecaea compactum, Fonsecaea pedrosoi, Phialophora verrucosa, Cladosporium carrionii, and Rhino-cladiella aquaspersa. The disease has a cosmopolitan distribution but predominates in rural, agricultural settings. Clinically, chromoblastomycosis is hallmarked by verrucous nodules at the site of fungal implantation. Treatment involves surgical excision of the affected area, antimycotic agents, physical modalities such as temperature manipulation, or a combination of these.     

Utility of helical computed tomography to evaluate the invasion of actinomycetoma; a report of 21 cases

BACKGROUND: Actinomycetoma is a chronic infection caused by several aerobic actinomycetes; it is a relatively frequent condition in tropical countries like Mexico. It is important to be aware of the extension and depth of the disease (bone and visceral) to make the prognosis and select treatment. OBJECTIVES: Our objective was to evaluate actinomycetomas using helical computed tomography (HCT) as well as its three-dimensional (3D) reconstruction. MATERIAL AND METHODS: Prospective study of clinically and microbiologically proven cases of actinomycetomas, all of them recently diagnosed and untreated or unresponsive to various treatments. All patients underwent simple and contrast HCT with various helical slices of the involved zones. Then three-dimensional reconstructions on the sagittal and coronal planes were made. RESULTS: Twenty-one patients with actinomycetomas were included, 19 males and two females, with a mean age of 35.5 years and mean duration of disease of 4.1 years. The disease was located in the lower limbs in 81%, and in the upper limbs and trunk in 19%. Twenty of the 21 cases were caused by Nocardia brasiliensis and one by Actinomadura madurae. In all patients the disease was localized to the skin and subcutaneous tissue; 76.2% had muscular involvement; 23.8% visceral involvement; 9.5% had bone involvement and 9.5% vascular involvement. The affected area was determined in each case. CONCLUSIONS: HCT provides precise information about the grade of invasion at diverse levels such as visceral, muscular and vascular systems, and the calculation of the affected area.     

Does the proximity of an amputation, length of time between foot ulcer development and amputation, or glycemic control at the time of amputation affect the mortality rate of people with diabetes who undergo an amputation?

OBJECTIVES: The main purpose of the authors' research was to compare the 3- and 5-year mortality rates of diabetic patients who have undergone a lower-extremity amputation, evaluating the proximity of the amputation in 3 groups (toe and foot amputation, BKA, and AKA), the timing of the amputation (within or after 2 years of the onset of the foot ulcer), and the effect of glycemic control at the time of amputation, regardless of the level of amputation. METHODS AND DESIGN: The subjects were 80 male inpatients at Illiana Veterans Health Care System who had diabetes, diabetic neuropathy, peripheral vascular disease, and a diabetic foot ulcer that resulted in an amputation. Of the 80 patients, 29 had a toe amputation, 30 had a BKA, and 21 had an AKA. The mean age in all 3 groups was 68.5 years+/-7.2 years. The authors used the hemoglobin A1C (Hgb A1C) level to assess glycemic control. MAIN OUTCOME MEASURES: Several chi tests were used to compare the 3- and 5-year mortality rates among the amputation groups. An Hgb A1C level of 8% or less defined good glycemic control; an Hgb A1C level of more than 8% defined poor glycemic control. A chi test was used to compare glycemic control, the level of amputation, and the mortality rate. A chi test was also used to evaluate the length of time between ulcer formation and amputation, the level of amputation, and the mortality. All statistics were done using SPSS 10.0 student version. CONCLUSIONS: The research revealed a statistically significant difference in mortality among the 3 groups at 3 and 5 years. No statistically significant difference in mortality in patients with good glycemic control and patients with bad glycemic control was found. At 3 years, a statistically significant difference in mortality existed between patients who had an amputation within 2 years of ulcer formation and those who had an amputation after 2 years. At 5 years, no statistically significant difference existed between these 2 groups.     

Botryomycosis. A bacterial cause of mycetoma.

Botryomycosis is a chronic, granulomatous, bacterial infection in which grains are produced. Clinically, it may not be distinguished from a mycetoma of fungal origin. A case is reported in which the causative organisms were Staphylococcus aureus and Pseudomonas aeruginosa.     

鳞状细胞癌和麻风慢性下肢溃疡

目的：进一步了解麻风下肢溃疡癌变的流行病学情况、临床特征及其治疗预后，为溃疡恶变的预防，早期发现和合理治疗提供参考。方法：对1980年6月－2001年9月期间由全省各地转到我院作住院治疗且有完整病历的麻风下肢溃疡癌变病例作回顾性分析，并对治疗结果作进一步追踪观察。结果：共收治21例，男15例，女6例；年龄48－71岁（平均59．1岁）溃疡病程8－30年（平均16．2年）；16例溃疡位于足底，4例位于小腿下部，1例位于踝部。病理分级：1级16例，2级5例；1993年10月以前的病例全部作了大腿截肢，此后的病例全部作了小腿截肢。3例失访；得到随访的18例中，截止2001年9月术后已平均存活37．7个月；已有9例死亡，大部分死亡术后1年左右。有10例确定有肿瘤转移，2例在局部，4例腹股沟淋巴结转移，4例发生远处转移。结论：溃疡病期在10年以上者是溃疡癌变的高发人群。早期诊断和治疗是提高存活期的关键     

Recurrent Scedosporium apiospermum mycetoma successfully treated by surgical excision and voriconazole

Scedosporium apiospermum is an emerging opportunistic fungus that can cause localized infection in healthy hosts or severe disseminated disease in immunocompromised hosts. Most cases are reported in Western Europe, Australia, and North America. We report a 52-year-old immunocompetent Taiwanese woman who presented with a 6-year history of recurrent asymptomatic papulonodular lesions on her right foot after minor trauma. Deep fungal infection caused by Scedosporium sp. was diagnosed after a skin biopsy with fungal culture of the skin specimen. She underwent two surgical excisions, each followed by a 4-month course of oral itraconazole and intralesional injections of amphotericin B as well, but similar lesions recurred at the same location 1 year later. She had another surgical excision and the pathological findings showed mycetoma. The fungus was identified as S. apiospermum by PCR assay of fungal culture specimen using the internal transcriber spacers (ITS1, similarity 99.4%; ITS2, similarity 100%) and the D1–D2 (similarity 99.0%) regions of the ribosomal operon. After 4 months of oral voriconazole (400 mg/day), no recurrence was noted in the subsequent 2 years.     

Deep fungal and higher bacterial skin infections in Thailand: clinical manifestations and treatment regimens

BACKGROUND: Deep fungal and higher bacterial skin infections occur fairly frequently in Thailand. METHODS: Cases with a provisional diagnosis of deep fungal and higher bacterial infections were prospectively collected from 1994 to 1997 in the Granuloma Clinic, Department of Dermatology, Faculty of Medicine, Siriraj Hospital, Mahidol University, Bangkok, Thailand. Demographic data, clinical manifestations, causative organisms, histologic features, treatment, and outcome were investigated. RESULTS: The total cases in a 4-year period numbered 27. The male to female ratio was approximately 1:1. Mycetoma was most common, followed by chromoblastomycosis. Actinomycetoma was similar in incidence to eumycetoma. The only causative organism that could be identified among the mycetoma cases was Cladosporium carrionii, which caused mycetoma of the buttock of an aplastic anemia patient at the site of bone marrow aspiration. Surgical treatment was recommended for eumycetoma. Chromoblastomycosis was caused by C. carrionii and F. compactum and responded well with itraconazole orally. Mycotic abscesses were found in four cases, basidiobolomycosis in two cases, and cutaneous nocardiosis in one case. Cotrimoxazole was recommended in the treatment of actinomycetoma, cutaneous nocardiosis, and basidiobolomycosis. CONCLUSIONS: Localized, chronic, slow, progressive, and usually asymptomatic were the main cutaneous manifestations of deep fungal and higher bacterial skin infections. A skin biopsy for histologic study and culture identification should be performed in every suspected case. The causative organisms were found in the histologic sections of every case, but only about one-third were found by culture.     

SMALL GRAIN MYCETOMAS IN THE TROPICS

Biopsy specimens from 159 patients with mycetoma filed in the departments of pathology of four medical colleges in Tamil Nadu, India, were examined histologically; small grain mycetoma due to Nocardia species was found in 27 cases. In addition, 17 clinically diagnosed cases of mycetoma without the characteristic discharging “granules” were investigated for the presence of Nocardia spp. by paraffin baiting and Nocardia spp. were isolated from nine of them. Of these, five were Nocardia asteroides, one was N. brasiliensis and three were identified as Nocardia spp. The classification and geographic distribution of Nocardia spp. are discussed.     

Cutaneous nocardiosis developing around gravel inserted during a traffic injury

A 65-year-old Japanese woman with nocardial mycetoma which developed five years after a compound bone fracture of the left foot due to a traffic accident was reported. During this accident, two small pieces of gravel had entered her foot. Nocardia asteroides, which had been attached to the gravel, was probably inserted 5 years previously during the trauma. The lesion did not respond to oral minocycline and trimethoprim-sulfamethoxazole and was finally removed surgically; she has been free from recurrences for one year, of follow-up.     

Actinomycotic mycetoma due to Nocardia brasiliensis in a case of leprosy

Various bacterial and fungal infections associated with non-healing ulcers in cases of leprosy have been reported (G Ebenzer et al, 2000, Rama Ramani et al, 1990). There are no reports of mycetoma associated with leprosy patients in the literature. We report here a case of actinomycotic mycetoma due to Nocardia brasiliensis associated with the non-healing plantar ulcer of a leprosy patient.     

Actinomycetoma and advances in its treatment

Actinomycetoma is a chronic subcutaneous infection caused by aerobic branching actinomycetes. Its clinical features are firm tumefaction of the affected site and the presence of abscesses, nodules, and sinuses that drain a seropurulent exudate containing filamenting granules. The disease is caused by inoculation of the infectious agent through minor trauma in susceptible individuals. Nocardia brasiliensis, Actinomadura madurae, and Streptomyces somaliensis are among the most frequent agents in the Americas. Cellular and humoral immunity have been studied in animal models. Standard therapy for uncomplicated cases is sulfamethoxazole-trimethoprim given for many months. Bone involvement, disseminated cases, and special locations require combined treatment with amikacin and sulfamethoxazole-trimethoprim. Isolated reports include the addition of other antibiotics such as rifampicin, imipenem, or meropenem. When needed, other aminoglycosides can be used. Amoxicillin-clavulanic acid is indicated in specific cases as alternative treatment. Oxazolidinone antibiotics, such as linezolid and other similar compounds (DA-7218 and DA-7157), have been studied in experimental infections in animal models as well as in vitro and ex vivo, with encouraging results.