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PubMedDate not supplied

Mycetoma caused by a new red grain mycetoma agent in two members of a family.

An 18-year-old woman from rural West Bengal was affected with mycetoma involving her neck, back, and chest. After an interval of eight years, her younger brother developed mycetoma on his left arm. No history of trauma or immune deficiency was present in either case. By microscopic examination of sinus-discharged materials from both the cases, identical rusty red, hard grains were demonstrated. Soluble red pigment-producing colonies grew in Sabouraud dextrose-agar medium. Isolates were positive for casein hydrolysis and negative for hydrolysis test of xanthine, hypoxanthine, tyrosine, and nitrate reduction. Thus it differed from the only known red grain mycetoma agent, Actinomadura pelletieri and was provisionally identified as Actinomadura vinacea. Familial affection in mycetoma, that too caused by a new agent, is reported here for its uniqueness.

Adolescent↗
PubMed1997

Criteria for the diagnosis of sinus mycetoma.

Recent improvements in the treatment options for fungal disease of the sinuses make rapid distinction between the syndromes of invasive and noninvasive sinusitis necessary. On the basis of the extensive review of the literature detailed here, we developed case-finding criteria for the noninvasive syndrome of sinus mycetoma. Using these criteria, we identified 20 cases of sinus mycetoma in the medical literature and compared findings in these patients to those of five patients with mycetoma evaluated in our clinics. Like those in the literature, our patients appeared immunocompetent and were often first seen for evaluation of symptoms other than those usually associated with sinusitis. Two were first seen with a new-onset seizure disorder and one with nasal obstruction alone. Patients from both groups more commonly had mycetoma in their maxillary sinuses, and fungus failed to grow from the cheesy, grey-green, hyphae-rich material removed at the time of surgery. Clinical features in our five patients, which are not noted in published reports of sinus mycetoma, included frequent atopy, nasal polyps, calcification within the sinus on computed tomography, and an excellent response to surgical treatment. Serendipitously, one patient had both allergic fungal sinusitis and a mycetoma in the same sinus. On the basis of this experience, we have modified our case-finding criteria into proposed diagnostic criteria for sinus mycetoma. The elements of these criteria are designed to: (1) exclude patients with invasive fungal sinusitis and (2) differentiate sinus mycetoma from other forms of noninvasive fungal sinusitis on the basis of specific histopathologic findings. This study and the criteria presented reflect our view that sinus mycetoma represents a distinct but not necessarily isolated element in the spectrum of noninvasive fungal disease of the sinuses.

Adult↗
PubMed2000

Lateral cavity wall thickening as an early radiographic sign of mycetoma formation.

The sensitivity of chest radiography for the early detection of mycetoma formation within fibrotic cavities is poor. The purpose of this study was to determine the predictive value of the secondary sign of lateral cavity wall thickening for the detection of a radiographically occult mycetoma. The chest radiographs and CT scans of 70 patients who had a total of 109 fibrotic cavities on CT were reviewed by two observers. Dimensions of the cavity, mycetoma, and cavity wall thickness on chest radiography and CT scans were recorded. Mycetomas were visible in 41 of 99 cavities on chest radiographs and in 61 of 109 cavities on CT. Using CT as the gold standard for detecting the presence of mycetomas, the sensitivity of chest radiography for the presence of a mycetoma was 62 % and the specificity 94 %, and the positive and negative predictive values were 93 and 66 %, respectively. On logistic regression analysis, lateral wall thickness on chest radiography was predictive of the presence of a mycetoma (p < 0.0005) independent of other radiographic features. In patients with chronic fibrocavitary disease on chest radiography, the presence of lateral wall thickening is highly suggestive of an underlying mycetoma.

Aspergillosis↗
PubMed2003

[Mycetoma: 130 cases].

BACKGROUND: Mycetoma is a pathological process in which eumycotic (fungal) or actinomycotic causative agents from exogenous source produce grains. It follows penetrating injury inoculating soil organisms, occurring preferentially in rural areas usually among labourers who work barefoot. Mycetoma is a localized chronic, and deforming infectious disease of subcutaneous tissues, skin and bones. We report 130 cases of mycetoma in Senegal from 1983 to 2000. PATIENTS AND METHODS: There were 130 patients with mycetoma. Clinical diagnosis of mycetoma was based on open tract sinuses, tumefaction or discharge of grain. Diagnosis confirmation was based on mycology and histology. An X-ray was preformed to detect bone lesions. Treatment was medical for actinomycetoma and surgical for eumycetoma. RESULTS: We observed 76 actinomycetoma and 54 eumycetoma (Sex ratio M/F=6.6; mean age=34.7 +/- 14.8 years). The mean duration before the first medical evaluation was 4.8 +/- 5.6 years. Actinomycetoma was due to Actinomadura pelletieri, (54 cases), Actinomadura madurae (17 cases) and Streptomyces somaliensis (5 cases). Eumycetoma was due to Madurella mycetomatis (38 cases), Leptospahria senegalensis (9 cases), Pseudoallescheria boydii (6 cases) and Rhinoclediella atrovirens (1 case). Clinical inflammatory features significantly associated with actinomyces (p<0.001 OR=2.64) were predominant (85 cases). Tumoral and cystic features were found in the others forms. Lesions were located on the foot in 81 patients. Bone lesions, depending on the duration, were observed in 68 patients. Neurological damage occurred in 3 patients with dorsolumbar actinomycetoma. Sixty-six patients with actinomycetoma were cured by medical treatment. DISCUSSION: The 130 cases of mycetoma were remarkable by the long duration of the disease before the first medical evaluation. Pain and tumor were the two main symptoms which brought the patients to the hospital and had appeared after 5 years duration and the predominance of actinomadura pelletieri actinomycetoma was responsible for 41.3 p. 100 of our cases. In Niger and Mauritania, mycetoma were actinomycetoma in respectively 71.2 p. 100 and 25 p. 100 of cases. The geographic distribution of pathogenic mycetoma agents was determined by the annual rainfall. Distinction between eumycetoma and actinomycetoma is very important for the treatment.

Adolescent↗
PubMed2002

Clinical presentation of pulmonary mycetoma in HIV-infected patients.

STUDY OBJECTIVE: s: Although pulmonary mycetoma has been well-described in immunocompetent hosts, the only description in HIV-infected patients has been of 10 patients from our institution, from 1992 to 1995. To further investigate the impact of HIV status on the presentation and course of pulmonary mycetoma, we conducted a follow-up study. DESIGN: Retrospective review of all cases of pulmonary mycetoma at Bellevue Hospital from 1992 to 1999. SETTING: Patients were evaluated on the inpatient chest service and in the outpatient chest and HIV clinics of Bellevue Hospital in New York City. PATIENTS: We identified 74 patients with pulmonary mycetoma; 20 of them were HIV-infected (27%). INTERVENTIONS: The 20 HIV-infected patients were treated with antiretroviral and/or antifungal therapy. MEASUREMENTS AND RESULTS: Predisposing diseases were pulmonary tuberculosis (TB), Pneumocystis carinii pneumonia (PCP), or both TB and PCP. Seventeen patients had a CD4+ cell count of < 100 cells/ micro L at presentation. Hemoptysis was present in 13 patients, but was massive in only 1 patient. Cough was common. Of the 18 patients for whom follow-up was available, 11 received antifungal treatment and 7 were observed without therapy. Six patients received both antiretroviral and antifungal therapy. Disease progression occurred in 50%. Only five patients exhibited radiographic or clinical improvement. All five were treated with both antiretroviral and antifungal therapy. CONCLUSIONS: PCP is a risk factor for pulmonary mycetoma in the HIV-infected individual. HIV-infected patients with mycetomas have a significant rate of disease progression, although they rarely have life-threatening hemoptysis. A combination of antifungal and antiretroviral therapy may improve the clinical outcome in HIV-infected patients with pulmonary mycetoma.

AIDS-Related Opportunistic Infections↗
PubMedDate not supplied

[Microsporum canis mycetoma of the scalp].

BACKGROUND: Mycetoma is a chronic subcutaneous tumefaction with presence of grains or granules. Etiological agents include bacteria or filamentous fungi. Mycetoma due to dermatophytes is uncommon, mainly occurring in Africa. To our knowledge, no case has been reported in the West Indies. Only two observations of Micosporum canis mycetoma in humans have been reported in the literature. We report a third case of mycetoma of the scalp caused by this fungus. CASE REPORT: A 22-year-old woman from Martinique, French West Indies, presented with an indolent tumefaction of the scalp evolving over five years. She had mental retardation due to congenital adrenal hyperplasia with 21-hydroxylase deficiency. The lesion was extracted surgically. Pathology and mycology examinations showed features of Microsporum canis mycetoma. Two months later, the scalp lesion recurred and the patient was treated with griseofulvin after surgical extraction. DISCUSSION: Mycetoma due to dermatophytes is very uncommon, mainly observed on the scalp and nape of the neck. A history of a skin lesion is frequent, leading to transcutaneous penetration of the fungus and mycetoma formation. Several dermatophyte species have been identified as causal agents (Microsporum ferrugineum, Trichophyton rubrum, Trichophyton verrucosum, Trichophyton mentagrophytes, Microsporum audouinii, Microsporum langeronii). Microsporum canis is rarely demonstrated in humans: two cases in children in Africa and Australia. Our observation was similar to the two cases in the literature: indolent and mobile tumefaction of the scalp, in a child or young adult, suggestive of lipoma or epidermal cyst, with excision leading to diagnosis. Association with tinea capitis and skin or nail involvement can also be observed.

Adult↗
PubMed1980

[Mycetoma and premycetomatous lesions in Uruguay].

Five cases of mycetomas and 5 "premycetomatous' lesions have been recorded in Uruguay. Two mycetomas were produced by fungi and three by nocardias. The casual fungi were identified as Scedosporium apoispermum in one case and Rubromadurella langeroni in the other. Nocardia asteroides was isolated from 2 cases and no cultures were obtained from the remaining one. Three mycetomas were diagnosed in an early stage of development and easy cures were obtained through medical or surgical procedures. In the other two cases the diagnosis was made in a late stage and the final result was amputation. A new clinical form of mycetoma called "lymphangitic' is proposed after describing a patient with mycetoma of the forearm and arm by S. apiospermum. Premycetomatous lesions included 3 cases of lymphangitic nocardiasis of the arm by N. brasiliensis, an abscess of the shoulder by the same species and an arthritis of the ankle by N. asteroides. All were cured after short periods of therapy. The usual early diagnosis of mycetomas and related diseases un Uruguay is considered to be the result of a prompt medical consultation due mainly to the abscence of important geographical obstacles in the Country.

Adult↗
PubMed2004

Serum immunoglobulins and IgG subclass levels in sinus mycetoma.

OBJECTIVE: To study the immunity of patients with sinus mycetoma by measuring serum immunoglobulins and IgG subclass levels. STUDY DESIGN AND SETTING: Thirty patients with sinus mycetoma, 71 ordinary chronic rhinosinusitis patients without nasal polyps, and 77 ordinary chronic rhinosinusitis patients with nasal polyps were collected. In all subjects, the levels of total serum immunoglobulins and IgG subclasses were measured during surgical treatment. RESULTS: Among 30 patients with sinus mycetoma, decreased IgA level was found in 1 patient, and decreased IgM level was found in another 2 patients. There were no significant differences in the levels of total serum immunoglobulins or IgG subclasses between patients with sinus mycetoma and ordinary chronic rhinosinusitis patients with or without nasal polyps. CONCLUSION: Immunologic defects cannot be detected in patients with sinus mycetoma by measuring levels of total serum immunoglobulins or IgG subclasses. SIGNIFICANCE: Immunoglobulin deficiency may not play an important role in the pathogenesis of sinus mycetoma.

Adolescent↗
PubMedDate not supplied

Fine needle aspiration cytology of mycetoma.

OBJECTIVE: To describe fine needle aspiration cytology of mycetoma and determine its usefulness in diagnosis. STUDY DESIGN: The study group consisted of 14 patients with different types of mycetoma lesions, which were aspirated. Smears were reviewed without knowing the type of mycetoma, and the findings were compared with those observed in histologic sections. RESULTS: In mycetoma, the causative organisms have a distinct appearance on cytologic smears. They are surrounded and infiltrated by neutrophils in a background of polymorphous, inflammatory cells consisting of neutrophils, histiocytes, lymphocytes, plasma cells, macrophages and foreign body giant cells. This allows differentiation from artifacts and inflammatory lesions caused by other bacteria and fungi. The distinction between eumycetoma and actinomycetoma in fine needle aspiration cytology was found to be as accurate as is histopathology when the grains were present. CONCLUSION: These results demonstrate that mycetoma can be accurately diagnosed by fine needle aspiration cytology. The technique is simple, inexpensive, rapid and sensitive. It can be used in the routine diagnosis of mycetoma, in epidemiologic surveys and in material collection.

Actinomycetales Infections↗
PubMed2026

A conserved grain-associated immunosuppressive niche in Sudanese patients with mycetoma.

Mycetoma is a neglected tropical disease caused by various bacterial and fungal pathogens that has a significant health impact across a broad geographically defined "mycetoma belt" spanning South America, Africa and Asia. Histologically, mycetoma is characterised by invasive and destructive granuloma development in the skin, deep tissues and bone, leading to tissue destruction, deformities and high morbidity. The presence of macroscopic, highly compacted pathogen microcolonies, or "grains," is a key diagnostic feature, and the formation of grains supports pathogen persistence and disease chronicity. However, there is a paucity of information on immune responses in mycetoma patients and on the relative importance of phylogeny and/or grains in establishing the local immune landscape. Here, we used spatial proteomics to examine the distribution of 43 immune-related proteins in surgical biopsies from 11 patients with mycetoma of bacterial (actinomycetoma; Actinomadura pelletierii and Streptomyces somaliensis; n&#x2009;=&#x2009;6) and fungal (eumycetoma; Madurella mycetomatis; n&#x2009;=&#x2009;5) origin. Using mixed-effects modelling, an exploratory analysis across species and pathogen classes revealed few significant differences in immune marker expression. In contrast, and independently of pathogen class, the cellular infiltrate closest to grain boundaries had higher per-cell expression of CD66b+ neutrophils, ARG1, and VISTA. The preferential accumulation of CD66b+ARG1+VISTA+ cells at grain boundaries was confirmed by quantitative immunofluorescence analysis. Hence, the local tissue microenvironment surrounding the mycetoma grain represents a specialised immunosuppressive niche, with parallels to the tumour microenvironment. Trial Registration The study was prospectively registered on ClinicalTrials.gov NCT04401969.

Adult↗
PubMed2004

Mycetoma of the hand.

OBJECTIVE: To describe the incidence and radiographic signs of hand infection in mycetoma. METHODS: A retrospective review was carried out from the radiographic records of 516 confirmed cases of mycetoma seen between January 1994 and October 2002 at the referred outpatient clinic of the Mycetoma Research Center, Soba Teaching Hospital, Khartoum University, Khartoum, Sudan. RESULTS: The hands were involved in 24 (4.6%) of the 516 patients. The wrist was involved in 9 (1.7%). Total involvement of the hand and wrist together was 33 (6.4%) of the 516 patients. Radiographic signs included fanning of the metacarpals, bone erosions, sclerosis, periostitis and soft tissue swelling. The most common sign was soft tissue swelling in 29 (88%) while bones were involved in 15 (45%) of the 33 patients. CONCLUSION: Hand infection in this series is less than previous reports. A wide range of radiographic signs is reported in hand mycetoma. This is the first detailed report of the radiographic findings in mycetoma of the hand. Early detection of mycetoma infection is important to avoid the need for amputation if diagnosis is delayed.

Adolescent↗
PubMedDate not supplied

Pathological fractures in mycetoma.

Pathological fractures in mycetoma are a rarity and only 1 patient was reported in the literature. We report an unexpectedly high incidence of pathological fractures in mycetoma seen at the Mycetoma Research Centre, Khartoum, Sudan over a 10-year period. Plain radiographs of 517 patients with confirmed mycetoma in various body sites were reviewed and correlated with the clinical presentation of the patients. Pathological fractures were detected in 12 patients (2.3%). The explanation of this high incidence is unclear. Pain-reducing agents produced by mycetoma may be the reason that these fractures were overlooked clinically.

Adolescent↗
PubMedDate not supplied

Fontana-positive grains in mycetoma caused by Microsporum canis.

We describe mycetoma caused by Microsporum canis occurring in a 9-year-old African-American girl. Pathologic evaluation showed a granulomatous dermatitis with numerous large fungal grains containing septate hyphae that were Fontana-Masson positive. Two cultures of pure grains grew M. canis. Mycetoma due to dermatophytes is very uncommon. The few instances reported have been pseudomycetoma (grains in the absence of sinus tracts). Our patient developed sinus tracts (true mycetoma). No prior reports were found of M. canis staining Fontana positive. Differentiation of dermatophyte-induced mycetoma from kerion is important because mycetomas require a combined approach including surgical debridement in addition to oral antifungal therapy.

Child↗
PubMed1993

Enzyme-linked immunosorbent assay for serological diagnosis of Nocardia brasiliensis and clinical correlation with mycetoma infections.

We previously identified three immunodominant antigens obtained from a Nocardia brasiliensis cell extract and recognized by sera from mycetoma patients (M. C. Salinas-Carmona, L. Vera, O. Welsh, and M. Rodríguez, Zentralbl. Bakteriol. 276:390-397, 1992). In the present work, we obtained a crude extract from a mass culture of N. brasiliensis HUJEG-1 and purified two immunodominant antigens, the 26- and 24-kDa proteins, by using simple physiochemical techniques. With these antigens, we developed a conventional solid-phase enzyme-linked immunosorbent assay and tested 30 serum samples from mycetoma patients, 29 from tuberculosis patients, 24 from a leprosy group, and 31 from healthy individuals. Our results show for the first time statistically significant differences in serology among these groups. All mycetoma patients with a positive culture for N. brasiliensis had absorbance values higher than 0.3. On the other hand, the mycobacterium-infected patients as well as the healthy individuals all had absorbance values below that level. Moreover, we found a close correlation between the clinical condition of the mycetoma patients and the anti-26- and anti-24-kDa protein antibody concentrations. We therefore propose the use of this assay in routine clinical laboratories to confirm the diagnosis of N. brasiliensis infection in human mycetoma cases. In addition, the possible application of this assay in the serodiagnosis of Nocardia asteroides infection is also discussed.

Antibodies, Bacterial↗
PubMed2002

Outcome of lung transplantation in patients with mycetomas.

BACKGROUND: Lung transplantation has become an acceptable treatment option for many end-stage lung diseases. Pulmonary mycetomas are found in patients with end-stage lung diseases, especially sarcoidosis. The clinical course and long-term outcome of these patients after transplantation remains unknown. METHODS: We reviewed retrospectively the pathology reports of the explanted lungs from all lung and heart-lung transplantations performed at our institution between January 20, 1992, and June 26, 2000. Patients were included in our study if mycetomas were present on the specimens. Information on transplant date and type, diagnosis, information on antifungal therapy and fungal infections pretransplant and posttransplant, and clinical course after transplantation was recorded. RESULTS: Mycetomas were present in 3.0% of transplant recipients (9 of 303 patients). The underlying pulmonary diagnoses were sarcoidosis (six patients), and emphysema, idiopathic pulmonary fibrosis, and pneumoconiosis (one patient each). Seven patients received bilateral lung transplants, one patient received a heart/lung transplant, and one patient received a single lung transplant. Aspergillus was isolated from culture in five patients pretransplant and from five patients posttransplant. Six patients received treatment with itraconazole, or IV or inhaled amphotericin B prior to transplantation. All patients who survived transplantation received posttransplant antifungal therapy. Four patients died in the first month after transplantation. Two patients died at 17 months and 24 months posttransplant, respectively; one patient received a second transplant 30 months later; and two patients are alive and free from fungal infections 17 months and 18 months, respectively, after transplantation. All of the medium-term survivors received lengthy therapy with inhaled and systemic amphotericin B and itraconazole before and after transplantation. CONCLUSIONS: Lung transplant recipients with mycetomas have significantly reduced posttransplant survival. Careful selection of patients and aggressive antifungal therapies before and after transplantation have led to improved outcomes in patients with mycetomas. Additional research is needed to define the best therapeutic strategy for these patients during transplantation.

Adult↗