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CASE REPORT

Madura’s Foot In Native Of The Philippines Emigrant In Northern Italy

*Azzoni Roberto, ”Cabitza Paolo

*MD, Assistant Professor in Orthopaedic Surgery,
“MD Professor in Orthopaedic Surgery and Chief of Orthopaedic Dept.,
Medical and Surgical Science Dept., State University of Milan.
20097 San Donato milanese, Milan 30, Morandi Street
, Italy.

Address for Correspondence:
Azzoni Roberto, MD
Orthopaedic Dept., Medical and Surgical Science Dept., State University of Milan.
20097 San Donato milanese, Milan, 30 Morandi Street I-Italy
Phone and fax number 0039-02-52774-632
E-mail roberto.azzoni@unimi.it paolo.cabitza@unimi.it
 

ABSTRACT

Clinical case about a man, emigrated from Philippines. The patient presented a single cutaneous lesion on a foot that was thick, swollen and blackish. There were no fistulas. He remenbers a traumatic implantation of wood splinters on the sole foot 10 years ago. Radiography, Magnetic Resonance, tomographic investigation were performed. The exceptional occurrence of the mycetoma in our countries and absence of clear infectious picture were the reason for an initial clinical misinterpretation as a benign neoplasm of soft tissues.We performed surgical removal of neoplasm, with histological examination.Histopathologic exam revealed an unexpected mass of fungal hyphae; diagnosis of Madura’s foot was confirmed. We started pharmacological therapy with “itracozanole”.Our case offers opportunity to stress need for clinical suspicion of fungal infection, considering increase of immigration by the countries with endemic mycetoma, and we must prepare to observe and treat many pathologies now unknown in our practice.
Key Words: mycetoma, eumycetoma, Madura foot, aspergillus fumigatus.

Introduction

Madura’s foot is a chronic skin infection due to various genera of fungi.  Usually is observed in tropical areas, particularly in India, Sudan, Senegal, Somalia, Mexico and Brazil where is endemic (1, 2, 3) but is exceptional in Europe. Actually the incidence of these cases is in continuous increase at our latitude because of movement of the world populations (4, 5). Recent data show that in England 2002-03 the 0,0001 p. 100 of hospital consultant episodes were for mycetoma (6). Today in Italy there is a considerable immigration from Northern and Central Africa, and from Far East, particularly Philippines. For this reason there is an increased observation of rare pathologies, but fungal infections are still uncommon. Unusual or atypical dermatological manifestations of infections can occur in persons who are immunocompromise by HIV or and in persons with prolonged stays in tropical and developing countries (7). This fungal infection is initially limited to the skin but may spread through the fascial planes up to contiguous structures as muscles and bone. Foot is the most common side of this infection (70%) (1). Gill (8) described this disease in the Indian Madura district in 1842, hence come the term Madura's foot. Madura’s foot, or mycetoma of the foot, is a slow growing infection, characterized by a thick and swollen lesion with sinuses draining a seropurulent and serosanguinous exudations containing granules with different colour (yellow, white, black, brown, or red) according to the type of eumycetes (4, 5). Etiologically two different groups of microrganisms are involved: bacteria belonging to the group of Actinomycetes (9, 10) and the true mycetes named Eumycetes. Eumycetes responsible for infection, (primarily saprophytic microorganisms), are found in the soil and on plant matter and are common in drier areas. Differently the Actinomycetes come from areas with relatively more rain. In any case the geographical distribution of clinical infection mirrors the pathogenic organism one (3).  These pathogenic agents as a result of the traumatic implantation of thorns, splinters, and other plant in healthy persons (2). For this reason foot is more common side of mycosis. Fungi that cause Eumycetoma include those with a white-to-yellow granules (like: Acremonium species, Aspergillus nidulans, Aspergillus flavus, Cylindrocarpon cyanescens, Cylindrocarpon destructans, Fusarium species, Neotestudina rosatii, Polycytella hominis, Polycytella boydii), and those with a black granules (like: Corynespora cassicola, Curvularia species, Exophiala jeanselmei, Geniculosporium, Leptosphaeria senegalensis, Leptosphaeria tompkinsii, Madurella grisea, Madurella mycetomatis, Phialophora verrucosa, Plenodomus auramii, Pyrenochaeta mackinnonii, Pyrenochaeta romeroi, Aspergillus fumigatus) (1, 2, 11, 12, 13).  Actinomycetes instead present white-yellow and brown-red grains. In the Philippines the most common fungal agents of Madura foot are Madurella grisea and actually we found a new species called Geniculosporium (11, 12, 14). Clinically Madura’s foot presents a single lesion that may persist for many years. It starts as a small hard painless lump under the skin and grows slowly involving underlying muscles and bone. The middle of the lesion caves in ulcerates and discharged pus, which contains grains. The cutaneous surface is scarred and pale. Often the plantar site of the foot has considerable deformity that makes difficult walking. Mycetoma may be asintomatic but usually itches or burns. And is common have a secondary infection in the same site.

Two opposing theories on etiology are reported in literature (15). The classical hypothesis is that a relatively limited list of typical agents causes mycetoma (16, 17). The second one is an unspecific response to the subcutaneous inoculation of a wider range of principally saprophytic agents (15, 18). Recent molecular data suggest that both suppositions are partly correct (18). Isn’t our intention to discuss here about etiology of mycetoma, but we think feasible both hypotheses too. Diagnostic cultural tests give certain identification to etiologic agent with recognition of the coloured grains. This data are obtained using a needle and syringe to extract material from a soft part of the lesion under the skin or by collecting pus, and making a cultural exam. Microscopical exams generally confirm surely the diagnosis and type of mycetoma. Occasionally is useful proceeding at a skin biopsy. Finally performing imaging studies like radiographs, tomographic scans, and with a magnetical resonance exams is possible demonstrate precisely the presence and extent of bone or organ involvement. Histological findings by biopsy specimen reveal, with hematoxylin-eosin stain, extensive granulation tissue containing grains of 0.2-0.5 mm. in diameter. With section Gomori methenamine stained grains are positives (1).

J.Orthopaedics 2005;2(6)e5

Case Report

Last year comes to our examination a man 40 years old, emigrated from the Philippines in Northern Italy. The patient presented a single cutaneous lesion that was thick, swollen and blackish, on the left foot, in the dorsal part between third and fourth toe. He had no pain and walk alterations. There weren't fistulas. He recalled at memory a traumatic implantation of wood splinters on the sole foot 10 years ago. An X-ray exam shows no involvement of bone (19). In any case was performed a magnetic resonance study (Fig. 1, 2, 3) that showed a tumefaction between the third and fourth metatarsal bones, beginning from the soft tissues. Multiple areas made it hyper-intense in T2 and STIR, tight near and corpuscle shaped. There weren't involvement of bone, and muscles but tight connection was present with tendons sheath. The exceptional occurrence of the mycetoma in our country and the lack of a clear clinical picture were the reason for an initial misinterpretation as a benign neoplasm of soft tissues. Therefore we performed a surgical removal of presumed neoplasm, with histological examination. During the surgical excision that was very easy to perform, the neoplasm looked like well defined, without any infiltration to neighbouring tissues. Only the subcutaneous tissues appeared suffering with swollen, infiltration had blacklish coloration. Histological examination was performed by hematoxylin and eosin, PAS, Gomori silver stain, and Grocott methenamine-silver stain. The histopathological pictures have revealed an unexpected mass of fungal hyphae with granulomatous inflammation near, and peripheric vallum of lymphocytes and histiocytes, and more peripheric areas of fibrosis (hematoxylin-eosin colouring) (Fig. 4). At PAS (Fig. 5), Gomori (Fig. 6) and Grocott colouring (Fig. 7), histopathological pictures have confirmed the fungal septate hyphae with black grains, typical of species Madurella grisea. Only with histologic result we perform a diagnosis of mycetoma due likely at Madurella grisea. The drugsterapy was started, ten days after surgery, with “itracozanole” (synthetic triazole antifungal agent that slows fungal cell growth by inhibiting cytochrome P-450-dependent synthesis of ergosterol, a vital component of fungal cell membranes). Posology of “itraconazole” is one capsule of 300 mg by mouth for at least three months (20, 21). A culture was performed with material collecting by needle and syringe from a soft part of the skin, unfortunately only ten days after surgery. The result of culture was positive for Madurella grisea, and reveals a secondary infection in the same site by Enterobacter cloacae. We have performed moreover a blood’s exam for searching antibodies against fungi, that was positive at  the same time for Aspergillus fumigatus (IgG title 1:80, with normal <1:40); in spite of previous therapy with “itraconazole”. And the patient was negative to the HIV test. Today patient has remission of local system, only the skin is more blackish, and few swollen (Fig. 8). Probably it would be the second case in Italy, quoted in literature (13), with normal immune status, treated successfully by surgical excision.



 

Discussion:

Mycetoma is a chronic granulomatous infection with starting-point of the skin with always involvement of subcutaneous tissues and often fascias, muscles, and occasionally bones and adjacent organs. The triad of symptoms that characterizes it: tumefaction, sinus tracts, and grain extrusion (9). Mycetoma is endemic disease around the Tropic of Cancer, in tropical and subtropical countries and temperate regions (2). In Europe, and particularly in Italy, this case is only rare report actually (5, 9, 22). Our case is still therefore a rare report, particularly for the atypical clinical picture till from the beginning. It presented a tumefaction, swollen, and blackish skin, but no present sinus tracts, and grain extrusion, characteristic of exogenous infectious agent likes the literature reports. Actually performing imaging exams (like radiographs, tomography scan, and magnetic resonance study) is useless for a precise diagnosis. The provenance of patient wasn’t from a country where mycetoma is endemic; usually it is useful element for the diagnosis. Only one element must been suspect: the anamnesis presented a traumatic implantation of wood splinters on the sole foot 10 years ago and the growth of tumefaction was been very slow. The poverty of symptoms and the atypical clinical picture were the reason for an initial clinical misinterpretation as a neoplasm of soft tissues, and only after histological exam correct diagnosis was performed. Our case offers the further opportunity to stress the necessity of a clinical suspicion about the fungal infection, considering the increase of immigration in Europe by the countries with endemic mycetoma. When we observe a tumefaction on the foot in a patient immigrated from a tropical, and subtropical countries, where frequently this pathology is endemic, and barefoot walking is very common; we must suspect a possibility of maduramycosis. Therefore we must perform any exam to make a correct diagnosis, for a precocious start of the right therapy as soon as possible. Today immigration to occidental countries is in continuous increase, and we must prepare ourself to observe and treat much pathology now unknown in our practice.

References:

  1. Turiansky G. Eumycetoma (Fungal Mycetoma). Www.eMedecine.com (>Specialities>Dermatology>Fungal Infections). 2002.

  2. Welsh O, Vera-Cabrera L, Salinas-Carmona MC. Mycetoma.www.eMedecine.com. (>Specialities>Dermatology>Fungal Infections). 2003.

  3. Dieng MT. Sy MH. Diop BM. Niang SO. Ndiaye B. Mycetoma: 130 cases. Ann. Dermatol. Vener. 130 (1): 16-23, 2003.

  4. Ten Broeke R, Walenkamp G.” The Madura foot: an innocent foot mycosis“? Acta Orthop. Belg. 64(2): 242-248, 1998.

  5. Verdolini R, Amerio P, Bugatti L, Manso E, Castaldi I, Brancorsini D, Nicolini M, Filosa G, Giangioacomi M. Madura’s foot: report of a case caused by Madurella mycetomatis. Eur. J. Dermatology 10(8): 627-629, 2000..

  6. Hospital Episode Statistics, Department of Health, England 2002-03.

  7. Lucchina LC, Wilson ME, Drake LA. Dermatology and the recently returned traveller: infectious diseases with dermatologic manifestations. Int. J. Dermatol. 36:167-181, 1997.

  8. Gill (1842), Indian Naval Medical reports. Quoted by Ghosh LM, et al. Madura foot (Mycetoma). Indian Medical Gazette. 85: 288-291, 1950.

  9. Rogopoulos D, Mavridou M, Nicolaidou E, Christofidou E, Antoniou C, Stratigos A, Katsambas A. Mycetoma due to actinomycetes: a rare entity in Europe. Int. J. Dermatology.  39(7): 557-558, 2000.

  10. Feasel A, Tschen J. A chronic draining plaque on the foot. Arch. Dermatology 2002; 138(10): 1371-1373.

  11. Reyes AC, Tangco AF, Punsalang AP Jr. Maduromycosis (maduromycotic mycetoma) in the Philippines caused by Madurella grisea. Southeast Asian J. Trop. Med. Public Health.  2(1):17-21, 1971.

  12. Suzuki Y, Udagawa S, Wakita H, Yamada N, Ichikawa H, Furukawa F, Takikawa M. Subcutaneous phaehyphomycosis caused by Geniculosporium species; a new fungal pathogen. Br. J. Dermatol. 138 (2): 346-349, 1998.

  13. Romano C, Miracco C. Primary cutaneous aspergillosis in an immunocompetent patient. Mycoses 46:56-59, 2002.

  14. Severo LC, Vetoratto G, Oliveira F, Londero AT. Eumycetoma by Madurella grisea: Report of the first case observed in the southern brazilian region. Rev. Inst. Med. trop. S. Paulo. 41(2): 139-142, 1999.

  15. De Hoog GS. Adelmann D. Ahmed AOA. Van Belkum A. Phylogeny and typification of Madurella mycetomatis, with a comparison of other agents of eumycetoma. Mycoses. 47: 121-130, 2004.

  16. De Hoog GS. Guarro J. Gené J. Figueras MJ. Atlas of clinical fungi. 2nd edn. Centraalbureau voor Schimmelcultures/Universitat Rovira i Virgili, Utrecht/Reus.  1126 p.. 2000.

  17. Rippon JW. Medical Mycology. The Pathogenic Fungi and the Pathogenic Actinomycetes. 3rd edn., Saunders, Philadelphia, 1988.

  18. Ahmed A. Adelmann D. Fahal A. Verbrugh H. van Belkum A. de Hoog GS. Environmental occurrence of Madurella mycetomatis, major agent of human eumycetoma in Sudan. J. Clin. Microbiol. 40: 1031-1036, 2002.

  19. Abd El Bagi ME. New radiographic classification of bone involvement in pedal mycetoma. Am. J. Roentg. 180(3): 665-668, 2003.

  20. Paugam A, Tourte-Schaefer C, Keita A, Chemla N, Chevrot A. Clinical cure of fungal madura foot with oral itraconazole. Cutis 60:191-193, 1997.

  21. Ahmed AOA, van de Sande WWJ, van Vianen W, van Belkum A, Fahal AH, Verbrugh HA, Bakker-Woudenberg IAJM. In vitro susceptibilities of Madurella mycetomatis to itraconazole and amphotericin B assesed by a modified NCCLS method and a viability-based 2,3-Bis(2-Methoxy-4-Nitro-5-Sulfophenyl)-5-[(phenylamino)Carbonyl]-2H-Tetrazolium Hydroxide (XXT) assay. Antimicrob. Agents Chemother. 48(7): 2742-2746, 2004.

  22. Dragonetti L, Cabitza P. Mycetoma foot (Madura foot). [Italian]. Arch. Ortop. 82: 407-420, 1969.

 

 This is a peer reviewed paper 

Please cite as : Azzoni Roberto: Madura’s Foot In Native Of The Philippines Emigrant In Northern Italy

J.Orthopaedics 2005;2(6)e5

URL:
http://www.jortho.org/2005/2/6/e5

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