This document discusses a case of mycetoma that was misdiagnosed for over 25 years in a non-endemic area. A 65-year-old man in Morocco presented with a chronic right foot lesion that was repeatedly misdiagnosed. After amputation, histopathological examination revealed findings consistent with eumycotic mycetoma. The case highlights the diagnostic challenges of mycetoma in areas where the disease is not commonly seen and practitioners are unfamiliar with it.
4. Background
• Madura foot or mycetoma is a chronic
granulomatous disease of subcutaneous
tissue, that can progress to deeper structures
(muscles or bones).
• Caused by either fungi (Eumycetoma) or by
aerobic filamentous bacteria
(actinomycetoma)
5. Background
• Affects mostly lower extrimities of the body
esp. foot and leg
• The disease often occurs in tropical and
subtropical regions of the world called
“mycetoma belt”
• India, Mexico, Senegal and Sudan are most
affected countries.
6. Background
• In 2014, Buonfare et al.
• Reported 42 cases in Europe, through a
literature review
• Without travel history by affected patients.
7. Background
• Sudan seems to be the most endemic country.
• But, in temperate climate, cases of mycetoma
mainly as imported cases from immigrants.
• Mycetoma encounter in rural area and agriculture
workers.
8. Background
• In 2013, the WHO listed this diseases among
neglected tropical disease.
• Several fungi and bacteria as causative agents
• Unfortunately, the diagnosis of the disease and
identification of the etiological agents is very
challenging issue, esp. in non-endemic areas.
9. Background
• Madura foot evolving for more than 2 decades
• Madura foot disease escaped all diagnostic tools
Misdiagnosed as cancer and leading to amputation
The final diagnosis has been achieved by the
histopathological examination of the resected
specimen.
10. Case presentation
• A 65yrs old man referred for evaluation of the right
tumor diagnosed recently as Kaposi’s sarcoma.
• He was a shopkepper living in the town of fes and
did not report any trip to an endemic area of
mycetoma.
• He had a right foot chronic lesion for 25yrs with
several repeated histological biopsies revealing,
keloid scar, non specific inflammation or Kaposi’s
sarcoma.
11. On examination
• Chronic skin changes on the right foot and leg with
multiple scars and hard abscessed ulcerations on
the plantar face of the foot.
• No grain discharge
• Culture of abscess shows staphylococcus aureus.
12. X- ray of the right foot shows extensive destruction
of the tarsal, meta-tarsal and phalanges.
13. Case presentation
• Other radiological evaluation did not found
further lesions.
• The diagnosis of locally invasive Kaposi’s
Sarcoma was suspected
• A right trans-tibial amputation was done.
14. Histopathological findings
• On macroscopic evaluation, the leg measured
30x11cm,
• Foot measured 27x10cm
• The foot showed an indurated skin, some areas of
hard abscess without any discharges.
• The initial sampling from these lesions showed a
non specific inflammation without any tumoral
lesion.
15. Histopathological findings
• Bone was sent for decalcification by nitric
acid.
• After decalcification:- macroscopic
evaluation found a deep soft tissue and bone
destruction consisted of round cavities filled
of yellowish crumply material.
17. Histopathological findings
• 0n HES stained sections revealed several
multilobulated colonies surrounded by
granulomatous inflammation composed of plasma
cells, epitheloid cells, macrophages and some
multinucleated cells.
• The colonies had deeply basophilic outer layers
with branching filaments.
• Some colonies were fractured and had a pale
center.
21. Histopathological findings
• These histological finding were strongly consistent
with Eumycotic Mycetoma.
• Post-operative course was uneventfull and
discharged.
• Two months later, no sign of disease and prosthesis
was prescribed.
22. Discussion
• Mycetoma is one of the neglected infectious
disease that is endemic in tropical and subtropical
areas of the world.
• Affects poor and rural people and in usually to
farmers.
• Affects all age groups but common in 20- 40 years
men.
23. Discussion
• Common in young men is due to productive age
group in developing countries.
• The low prevalence in women could be due to
hormonal factors as in women take part in
agriculture work.
• In areas out of “mycetoma belt”as in Temperate
region- disease seen mainly in immigrants.
24. Discussion
• Also cases from indigenous of non endemic zone
without travel history have been reported.
• Health practitioners in these areas are not familiar
to the disease
• Cases were usually misdiagnose and mismanaged
leading to serious consequences.
25. Discussion
• This patients from Morocco, ( out of “mycetoma
belt”) where till now < 100 cases reported.
• More than 56 micro-organism ( fungi or bacteria)
are known to date to be linked mycetoma.
• Found in plants thorn or in the soil.
• People become infected by thorn prick or waking
barefoot.
26. Discussion
• Prevalence of causative varies in the world.
• In Sudan main causative agents are fungi
• In Latin America, Mexico- bacterial agents are
predominant.
• Common Actinomycetoma are: Actinomadura
madurae, Streptomyces somaliences,
Actinomadura pelletieri, Nocardia asteroids.
27. Discussion
• Eumycetoma – Madurella mycetomatis.
• Clinical presentation is similar (fungi or bacteria)
• However Actinomycetoma has more aggressive
course and invade deeper strctures earlier than
Eumycetoma.
28. Discussion
• Typical presentation of a classical triad:
Painless firm subcutaneous mass
Multiple sinus formation
Purulent or seropurulent discharge containing
grains.
29. Discussion
• Disease pursue a long course due to its:
Painless features
Lack of appropriate health information about
the disease
Its occurs in poorly educated patients
Misdiagnosis especially in non endemic regions
30. Discussion
• In this case, >20 years course, repeatedly
misdiagnosed, leading to leg amputation.
• Similarly, mycetoma cases have been reported in
Europe, with long course and subsequent
amputation.
31. Discussion
• The more challenging issue with eumycetoma is the
diagnosis in early stage of the disease.
• Becomes more challenging to identify causative
agent.
• Treatments depend on agents type, severity and
extension.
33. Discussion
• Culture methods are gold standard to identify
causative agents.
• However, it is time consuming and chance of
contamination.
• In this case, culture shows Staph. aureus as
contamination.
34. Discussion
• Skin test and serology could be used but not also
fully reliable.
• Currently molecular technics are only reliable
diagnostic tool to identify the exact species of the
causative agents.
• Drawback is high cost for developing countries
(endemic area ).
35. Discussion
• Histopathology is another diagnostic tool to
identify causative agents.
• Merit of pathology is to differentiate eumycetoma
from actinomycetoma.
• Drawback- cannot identify at species level is almost
impossible.
• Grains of causative agents can be obtained by
cotton swab from sinuses, by FNA, or by biopsy.
36. Discussion
• Deep seated grains provide more diagnostic
information.
• Macroscopic examination of grains do not provide
any specific diagnostic orientation.
• Eumycetoma- have black, white or yellow grains.
• Actinomycetoma have yellow, white, red or pink
grains.
37. Discussion
• Longstanding disease, discharge from sinuses is
scarce or completely inapparent due to extensive
fibrosis.
• Biopsy shows misleading features as non- specific
inflammation or mimic certain malignancies.
• Reactive fibroblast and histiocytes along with
fibrotic and hemorrhagic changes lead to think
pathologist about Kaposi Sarcoma.
38. Discussion
• Long course along with extension to adjacent
structure also lead to misdiagnosis as malignancies.
• Another case in Morocco, patients was diagnosed
with Kaposi sarcoma and given chemotherapy
before the correct diagnosis of mycetoma.
39. Discussion
• This patients also after several biopsies that shows
non-specific inflammation and concluded to
kaposi’s sarcoma invading bone structures that
justifying amputaion.
• Histopathological approach uses HES stain
combined with other special stains such as PAS,
Gram, ZN stain, Grocott stain etc.
40. Discussion
• With HES stain, grains represent colonies of
causative agents surrounded by granulomatous
inflammation comprising plasma cells, neutrophils,
macrophage and gaint cells.
• Colonies from actinomycetoma have different size,
round or multilobulated , with deeply stained
basophilic outer border and pale center.
41. Discussion
• Splendore-Hoeppli Phenomenon:
Eosinophilic hyaline like material surrounds the
colonies.
• Colonies may show fractured aspect.
• Filaments are thin and <1μm.
• Typically actinomycotic colonies are gram positive
and negative for PAS.
• Nocardia positive for ZN stain.
42. Discussion
• Histologically this case positive to PAS stain ruled
out Actinomycetoma.
• Colonies from Eumycetoma show several
histological overlapping appearance with
Actinomycetoma colonies but their filaments are
thicker, 2-6μm.
• Eumycetoma stain positive for PAS and negative for
gram or ZN stain.
43. Discussion
• Pathology also rule out any malignancy or specific
granulomatous inflammations such as TB.
• Treatment of mycetoma depends on causative
agents, either fungal or bacterial.
• Antifungal or antibacterial drugs along with
sometimes combined with surgery.
• Eumycetoma – Azole class of drug used.
44. Discussion
• Recurrences are frequent and compliance to
treatment seems difficult.
• For actinomycetoma, cotrimoxazole with amikacin
for weeks.
• Prognois better in Actinomycetoma compared to
Eumycetoma.
46. Discussion
• Despite of long term treatment and recurrences,
aggressive surgery is not the first line of treatment.
• Amputation are generally due to misdiagnosis and
longstanding disease spreads deeper structures.
• This patients should treat medically rather than
surgically.
47. Discussion
• There was misdiagnosis due to the fact that
clinicians were not familiar to Mycetoma in
Morroco as it not an endemic area of Mycetoma.
• So misdiagnosis and mismanagement are common
in non endemic regions.
48. Conclusion
• The case is from non- endemic area
• Evolve in two decades
• Misdiagnosis as an cancer
• Unnecessary and aggressive surgery.
• Diagnostic challenge of mycetoma in developing
countries and non- endemic areas.
• Clinician should aware of that in order to provide
early diagnosis and treatment.
49. TAKE HOME MESSAGE
• Misdiagnosis and mismanagement is common in non
endemic areas.
• Medical management is first line of therapy or along with
surgical debridement.
• Molecular technics are only reliable diagnostic tool to
identify exact species of causative agents.
• Histopathology is another tool that can aid to identify the
causative agent.
