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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.
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