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      A Rare Presentation of Concurrent Scedosporium apiospermum and Madurella grisea Eumycetoma in an Immunocompetent Host

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          Abstract

          Mycetoma is a disfiguring, chronic granulomatous infection which affects the skin and the underlying subcutaneous tissue. We present an atypical case of recurrent mycetoma without ulceration, in a 35-year-old immunocompetent male caused by Scedosporium apiospermum sensu stricto and Madurella grisea, occurring at two separate anatomical sites.

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          Molecular and phenotypic data supporting distinct species statuses for Scedosporium apiospermum and Pseudallescheria boydii and the proposed new species Scedosporium dehoogii.

          Based on the morphological, physiologic, and molecular (beta-tubulin gene) study of 141 isolates of the Pseudallescheria boydii species complex (including several synonyms) and relatives, the new species Scedosporium dehoogii is proposed. Scedosporium apiospermum and P. boydii are considered two different species and the new name Scedosporium boydii is proposed for the anamorph of the latter species. A summary of the key morphological and physiological features for distinguishing the species of Pseudallescheria/Scedosporium is provided.
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            Mycetoma

            A 46-year-old male, a grocer by occupation was referred from the orthopedics department for swelling of his right leg with discharge from overlying skin since 18 months. He was a known case of chronic osteomyelitis since 7 years. Cutaneous examination revealed diffuse swelling of right thigh and leg along with effusion of right knee joint. Also, there were multiple sinuses on right thigh and on upper one-third of right leg. Two sinuses were found discharging serosanguinous fluid along with black granules [Figure 1]. Multiple healed atrophic scars were noticed on right thigh. KOH preparation of the crushed granules showed septate branching phaeoid hyphae [Figure 2]. Culture on Sabouraud's dextrose agar was negative for maduramycosis. Radiograph of the right leg showed circumscribed osteolytic lesion in the metaphyseal region of right tibia [Figure 3]. Magnetic resonance imaging (MRI) of right leg showed well-defined, ovoid, mixed intensity lesion in the metaphyseal region of right upper one-third of tibia. The 'dot-in-circle' sign was visualized [Figure 4]. Lesional biopsy was noncontributory. Characteristic clinical morphology, microscopy of the granules, radiography, and MRI assisted in establishing the diagnosis of mycetoma. Figure 1 Multiple sinuses on right thigh and upper one-third of right leg. Two sinuses show discharging serosanguinous fluid along with black granules Figure 2 KOH mount of the crushed granules showing septate branching phaeoid hyphae Figure 3 Radiograph of right leg showing circumscribed osteolytic lesion in the metaphysical region of right tibia Figure 4 MRI of right leg showing well-defined ovoid mixed intensity lesion in the metaphyseal region of right upper one-third tibia with the 'dot-in-circle' sign (arrow) Mycetoma is a chronic granulomatous infection of the skin and subcutaneous tissues characterized by induration, abscess formation with draining sinuses. It is caused by both fungi (eumycetoma) and filamentous bacteria (actinomycetoma). It derives its name from Madurai, the place where it was first reported. The clinical features are the same irrespective of the causative agent.[1] Madurella mycetomatosis is one of the commonest causes of eumycetoma[2] and it is common among males who perform more outdoor activity. Usually follows injury such as a thorn prick or an injury resulting in ulceration. Similarly, mycetoma followed vehicular injury in our case. The foot is a common site of involvement followed by and extremities and perineum.[3] Involvement of the upper part of the leg in our case is in concert with the common sites of involvement. Confirmation of the diagnosis is by demonstration of the fungus and speciation by culture on Sabouraud's dextrose agar.[4] However, only fungal elements could be demonstrated in our case but failed to culture the fungus on Sabouraud's dextrose agar. The surface of the colony is heaped up, and radially folded with a glabrous to wooly texture. Its color varies from white to yellow-brown to dark gray or olive brown, whereas the reverse shows brown pigment. The causative fungus could not be established in our case as it could not be cultured. Nonetheless, black granules were found in the discharge overlying the sinuses, which is a characteristic clinical feature of eumycetoma. Bone involvement is a major complication in maduramycosis resulting in osteolytic lesions seen on radiography.[5] The upper end of tibia showed osteolytic lesions in our case. The recently described dot-in-circle sign on magnetic resonance imaging (MRI) is highly specific of maduramycosis.[6] Treatment of eumycetoma consists of antifungal drugs combined with surgery. Antifungal drugs such as itraconazole, posaconazole, ketoconazole, and terbinafine long are required to be administered for a long duration. Financial support and sponsorship Nil. Conflicts of interest There are no conflicts of interest.
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              Mycetoma.

              Mycetoma is a granulomatous infection affecting mainly the feet and lower extremities. It can be caused either by aerobic, branched actinomycetes or by eumycetes. Most cases are found in tropical and subtropical regions. The infection is usually produced by the introduction of the etiologic agents through minor wounds caused by thorns and wood splinters. Clinically the disease begins as small, firm nodules that can enlarge to form extensive lesions with fistulae and abscesses with pus containing granules of the causative microorganisms. Antimicrobials and surgery are used in the management of mycetoma. The actinomycetomas generally respond well to antimicrobials. For eumycetomas, surgery may be required. New therapeutic options for drug-resistant cases are discussed.
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                Author and article information

                Journal
                Case Report Pathol
                Case Report Pathol
                CRIM.PATHOLOGY
                Case Reports in Pathology
                Hindawi Publishing Corporation
                2090-6781
                2090-679X
                2012
                22 October 2012
                : 2012
                : 154201
                Affiliations
                1Department of Orthopaedic, Ealing Hospital NHS Trust, Uxbridge Road, Southall UB1 3HW, UK
                2HPA Mycology Reference Laboratory, Myrtle Road, Bristol BS2 8EL, UK
                Author notes

                Academic Editors: T. Batinac, C. A. Palmer, M. S. Patel, and P. Perrini

                Article
                10.1155/2012/154201
                3485492
                23133772
                1789bfb6-5574-4c06-bd3e-d8885ab85d0e
                Copyright © 2012 Vivek Gulati et al.

                This is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

                History
                : 17 September 2012
                : 9 October 2012
                Categories
                Case Report

                Pathology
                Pathology

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