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Background. Basidiobolomycosis is a rare subcutaneous mycosis, which can be mistaken for several other diseases, such as soft tissue tumors, lymphoma, or Buruli ulcer in the preulcerative stage. Microbiological confirmation by PCR for Basidiobolus ranarum and culture yield the most specific diagnosis, yet they are not widely available in endemic areas and with varying sensitivity. A combination of histopathological findings, namely, granulomatous inflammation with giant cells, septate hyphal fragments, and the Splendore-Hoeppli phenomenon, can confirm basidiobolomycosis in patients presenting with painless, hard induration of soft tissue. Case Presentations. We report on three patients misdiagnosed as suffering from Buruli ulcer, who did not respond to Buruli treatment. Histopathological review of the tissue sections from these patients suggests basidiobolomycosis. All patients had been lost to follow-up, and none received antifungal therapy. On visiting the patients at their homes, two were reported to have died of unknown causes. The third patient was found alive and well and had experienced local spontaneous healing. Conclusion. Basidiobolomycosis is a rare subcutaneous fungal disease mimicking preulcerative Buruli ulcer. We stress the importance of the early recognition by clinicians and pathologists of this treatable disease, so patients can timely receive antifungal therapy. [ABSTRACT FROM AUTHOR]

Background: Buruli ulcer (BU) is a neglected tropical disease caused by Mycobacterium ulcerans. Usually BU begins as a painless nodule, plaque or edema, ultimately developing into an ulcer. The high number of patients presenting with ulcers in an advanced stage is striking. Such late presentation will complicate treatment and have long-term disabilities as a consequence. The disease is mainly endemic in West Africa. The primary strategy for control of this disease is early detection using community village volunteers. Methodology/Principal Findings: In this retrospective, observational study, information regarding Buruli ulcer patients that reported to one of the four BU centers in Bénin between January 2008 and December 2010 was collected using the WHO/BU01 forms. Information used from these forms included general characteristics of the patient, the results of diagnostic tests, the presence of functional limitations at start of treatment, lesion size, patient delay and the referral system. The role of the different referral systems on the stage of disease at presentation in the hospital was analyzed by a logistic regression analysis. About a quarter of the patients (26.5%) were referred to the hospital by the community health volunteers. In our data set, patients referred to the hospital by community health volunteers appeared to be in an earlier stage of disease than patients referred by other methods, but after adjustment by the regression analysis for the health center, this effect could no longer be seen. The Polymerase Chain Reaction (PCR) for IS2404 positivity rate among patients referred by the community health volunteers was not systematically lower than in patients referred by other systems. Conclusions/Significance: This study clarifies the role played by community health volunteers in Bénin, and shows that they play an important role in the control of BU. Author Summary: Buruli ulcer (BU) is a neglected tropical disease caused by Mycobacterium ulcerans. Usually, the number of patients presenting with ulcers in an advanced stage is high. This complicates treatment and increases the risk of disabilities. The disease is endemic mainly in West Africa. The primary strategy for control is early detection using community village volunteers. In Bénin, data was collected to understand the role of the different referral systems on the stage of disease at presentation in the hospital and the diagnostic precision. About a quarter of the patients were referred to the hospital by the community health volunteers. Community health volunteers referred patients more frequently in an earlier stage of disease. The PCR confirmation rate among patients referred by the community health volunteers was not systematically lower than in patients referred by other systems. We found that community health volunteers played an important role in the referral system of BU patients in Bénin. This information is relevant for health care programs attempting to control BU but may also be relevant for health care programs working on other diseases in areas with restricted resources. [ABSTRACT FROM AUTHOR]

We identified risk factors for Buruli ulcer (BU) in Benin in an unmatched case-control study at the Centre Sanitaire et Nutritionnel Gbemoten in southern Benin. A total of 2,399 persons admitted from 1997 through 2003 and 1,444 unmatched patients with other conditions in 2002 were recruited. Adjusted odds ratios were determined for age, sex, place of residence, Mycobacterium bovis BCG vaccination at birth, type of water for domestic use, and occupation. Children <15 years of age and adults >49 years of age had a higher risk for BU. Use of unprotected water from swamps was associated with increased risk for BU; this association was strongest in adults >49 years of age. Sex was not a risk factor for BU. Our data showed that BU was mainly associated with age, place of residence, and water sources in all age groups. Risk for BU was higher in BCG-vaccinated patients ≥5 years of age. [ABSTRACT FROM AUTHOR]

Buruli ulcer is a recognized public health problem in West Africa. In Benin, from 1989 to 2001, the Centre Sanitaire et Nutritionnel Gbemoten (CSNG) treated <2,500 Buruli ulcer patients. From March 2000 to February 2001, field trips were conducted in the Zou and Atlantique regions. The choice of the 2 regions was based on the distance from CSNG and on villages with the highest number of patients treated at CSNG. A total of 66 (44.0%) of 150 former patients treated at CSNG were located in the visited villages. The recurrence rate of CSNG-treated patients after a follow-up period of up to 7 years was low (6.1%, 95% confidence interval [CI] 2.0-15.6). We attribute this low rate to the high quality of Buruli ulcer treatment at an accessible regional center (CSNG). The World Health Organization definition of a Buruli ulcer recurrent case should be revised to include a follow-up period >1 year. [ABSTRACT FROM AUTHOR]

Data from 1,700 patients living in southern Benin were collected at the Centre Sanitaire et Nutritionnel Gbemoten, Zagnanado, Benin, from 1997 through 2001. In the Zou region in 1999, Buruli ulcer (BU) had a higher detection rate (21.5/100,000) than leprosy (13.4/100,000) and tuberculosis (20.0/100,000). More than 13% of the patients had osteomyelitis, Delay in seeking treatment declined from 4 months in 1989 to 1 month in 2001, and median hospitalization time decreased from 9 months in 1989 to 1 month in 2001. This reduction is attributed, in part, to implementing an international cooperation program, creating a national BU program, and making advances in patient care. [ABSTRACT FROM AUTHOR]

We reviewed Buruli ulcer (BU) surveillance in Benin, using the World Health Organization BU02 form. We report results of reliable routine data collected on 2,598 new and recurrent cases from 2003 through 2005. [ABSTRACT FROM AUTHOR]

An otherwise healthy, afebrile, 13-year-old girl presented to a rural hospital in Benin with a small superficial ulcer of the skin of the right forearm and marked induration extending from the dorsum of the hand to the elbow, without regional lymphadenopathy. A playmate had bitten her one month previously. Clinical diagnosis of disseminated Mycobacterium ulcerans disease. Histopathological changes included extensive coagulation necrosis of subcutaneous tissue and fascia, dead fat cells, and clumps of acid-fast bacilli.

Background: Increased availability of Next Generation Sequencing (NGS) techniques allows, for the first time, to distinguish relapses from reinfections in patients with multiple Buruli ulcer (BU) episodes., Methodology: We compared the number and location of single nucleotide polymorphisms (SNPs) identified by genomic screening between four pairs of Mycobacterium ulcerans isolates collected at the time of first diagnosis and at recurrence, derived from a collection of almost 5000 well characterized clinical samples from one BU treatment center in Benin., Principal Findings: The findings suggest that after surgical treatment-without antibiotics-the second episodes were due to relapse rather than reinfection. Since specific antibiotics were introduced for the treatment of BU, the one patient with a culture available from both disease episodes had M. ulcerans isolates with a genomic distance of 20 SNPs, suggesting the patient was most likely reinfected rather than having a relapse., Conclusions: To our knowledge, this study is the first to study recurrences in M. ulcerans using NGS, and to identify exogenous reinfection as causing a recurrence of BU. The occurrence of reinfection highlights the contribution of ongoing exposure to M. ulcerans to disease recurrence, and has implications for vaccine development.

Invasive punch or incisional skin biopsy specimens are currently employed for the bacteriological confirmation of the clinical diagnosis of Buruli ulcer (BU), a cutaneous infectious disease caused by Mycobacterium ulcerans. The efficacy of fine-needle aspirates (FNA) using fine-gauge needles (23G by 25 mm) for the laboratory confirmation of BU was compared with that of skin tissue fragments obtained in parallel by excision or punch biopsy. In three BU treatment centers in Benin, both types of diagnostic material were obtained from 33 clinically suspected cases of BU and subjected to the same laboratory analyses: i.e., direct smear examination, IS2404 PCR, and in vitro culture. Twenty-three patients, demonstrating 17 ulcerative and 6 nonulcerative lesions, were positive by at least two tests and were therefore confirmed to have active BU. A total of 68 aspirates and 68 parallel tissue specimens were available from these confirmed patients. When comparing the sensitivities of the three confirmation tests between FNA and tissue specimens, the latter yielded more positive results, but only for PCR was this significant. When only nonulcerative BU lesions were considered, however, the sensitivities of the confirmation tests using FNA and tissue specimens were not significantly different. Our results show that the minimally invasive FNA technique offers enough sensitivity to be used for the diagnosis of BU in nonulcerative lesions.

We investigated the association between Buruli ulcer and HIV by comparing the HIV-1/2 seroprevalence in a series of 426 Buruli ulcer patients and a sample of 613 residents of southern Benin. The overall HIV prevalence was 2.6% (11/426) among patients and 0.3% among controls (2/613), giving an odds ratio for the association between HIV and Buruli ulcer of 8.1 (95% confidence interval = 1.8-75; P = 0.003).

Tissue specimens collected from patients with clinically suspected Buruli ulcer treated in two Buruli ulcer treatment centers in Benin between 1998 and 2004 were placed in semisolid transport medium and transported at ambient temperature for microbiological analysis at the Institute of Tropical Medicine in Antwerp, Belgium. The impact of the delay before microbiological analysis on primary culture of Mycobacterium ulcerans was investigated. The length of storage in semisolid transport medium varied from 6 days to 26 weeks. Of the 1,273 tissue fragments positive for M. ulcerans DNA by an IS2404-specific PCR, 576 (45.2%) yielded positive culture results. The sensitivity of direct smear examination was 64.6% (822/1,273 tissue fragments). The median time required to obtain a positive culture result was 11 weeks. Positive cultures were obtained even from samples kept for more than 2 months at ambient temperatures. Moreover, there was no reduction in the viability of M. ulcerans, as detected by culture, when specimens remained in semisolid transport medium for long periods of time (up to 26 weeks). We can conclude that the method with semisolid transport medium is very robust for clinical specimens from patients with Buruli ulcer that, due to circumstances, cannot be analyzed in a timely manner. This transport medium is thus very useful for the confirmation of a diagnosis of Buruli ulcer with specimens collected in the field.