A Case of Bilateral Painful Eumycetoma of the Feet Responding Excellently to Low Dose Itraconazole.

Respected Sir,

Mycetoma is a chronic localized infection involving the skin, subcutaneous tissues, and bone of the unilateral hand or feet, less commonly other body parts. This chronic localized suppurative infection is caused by various species of fungi (eumycotic) or filamentous bacteria (actinomycotic) and is characterized by the formation of grains within the tissue and discharge of the same to the surface through draining sinuses. This results in severe damage to the skin, subcutaneous tissues, and bones.

A 47-year-old housewife presented with multiple slowly progressive nodules over the dorsum of both feet for 4 years. The patient gave a history of walking barefooted and complained of pain while walking. She was withdrawn; complained of lack of sleep, lethargy, and feeling of guilt. On examination, a hyperpigmented swelling with minimal scaling was seen over the dorsum of both feet extending up to the toes. Multiple draining sinuses with a history of pus discharge were seen over both feet. Nearly all the toenails were dystrophic [Figure 1]. On palpation, there was no local tenderness and the lesion appeared to be firm. She was non-diabetic, non-HIV reactive, and otherwise, apparently healthy. We thought of mycetoma, cutaneous tuberculosis, atypical mycobacterial infection, chromoblastomycosis, and osteomyelitis.

Figure 1

Hyperpigmented swelling with multiple draining sinuses over the dorsum of both feet

Routine investigations including complete hemogram, chest X-ray, renal and liver function tests were within the normal limit [Figure 2]. Anterior–posterior (AP) and lateral views of the feet X-ray did not reveal any bony abnormality [Figures 3 and 4]. Grains could not be collected from the scanty pus discharge in spite of multiple attempts. Saline-soaked gauge dressings on the sinus tracts did not yield any grains either. A biopsy was taken including the whole of a sinus tract and sent for Gram staining, acid-fast staining, tuberculosis-polymerase chain reaction (TB-PCR), and hematoxylin–eosin (H and E) staining. Gram staining and acid-fast staining did not show any bacteria. TB-PCR result was also negative. H and E staining showed aggregates of eosinophilic material surrounded by mixed inflammatory cell infiltrate in the dermis and deeper tissues [Figure 5]. The eosinophilic aggregates were mycotic granules with collections of the fungus. Periodic acid Schiff (PAS) stain further revealed thick-walled fungal hyphae [Figure 6]. We could not perform any fungal culture due to lack of facility. Based on the clinical presentation and histopathology appearance, a diagnosis of bilateral eumycotic mycetoma of the feet was made and the patient was put on low-dose itraconazole 200 mg daily therapy. She showed dramatic improvement with the clearing of all lesions after 8 months of therapy. The patient was treated successfully with an antidepressant for pain and depressive symptoms. At 1.5-year post-therapy follow-up, the patient did not show any sign of recurrence [Figure 7].

Figure 2

Chest X-ray was within normal limit

Figure 3

Anterior–posterior and lateral views of the right feet

Figure 4

Anterior–posterior and lateral views of the left feet

Figure 5

Eosinophilic material surrounded by a mixed inflammatory infiltrate in the deep dermis

Figure 6

Periodic acid Schiff staining shows fungal hyphae

Figure 7

Follow-up picture after 1.5 years

Eumycetoma is caused by at least 23 fungal species, most commonly by Madurella mycetomatis.[1] Even though mycetoma is relatively endemic in this part of the country, eumycetoma is fairly uncommon in West Bengal.[2] H and E stain may show aggregates of septate and branched broad hyphae with neutrophils invading the area, which is surrounded by palisading histiocytes, in turn, surrounded by a mixed inflammatory cell infiltrate. Fungal culture remains the gold standard of diagnosis but, usually takes 4 to 6 weeks for a positive report.[1]

Our patient presented with the hallmarks of mycetoma: multiple swellings with draining sinuses and involvement of both of her feet. Bilateral presentation is a rare occurrence too for that matter in the world scenario.[3] Thus, in endemic areas, eumycetoma should be considered in the differential diagnosis of bilateral chronic subcutaneous lesions. No macroscopic grains were observed. This may happen in eumycotic mycetoma due to a lack of cementing substance as reported by Tomimori-Yamashita et al.[4] The clinical diagnosis was corroborated by the histopathological findings.

Itraconazole, a triazole antifungal drug, was released in the early 1990s and has become the most commonly used drug for the treatment of eumycetoma when and where it is affordable.[56] On extensive literature search, we found almost all cases of eumycetoma reported so far required combined surgical clearance and antifungal therapy. Only a few cases responded excellently to medical therapy that too to voriconazole and posaconazole. Complete cure in our case without any further recurrence to itraconazole is definitely exciting in this context. Welsh et al.[7] and Elkheir et al.[8] have recommended itraconazole at a dose of 200 to 400 mg daily for eumycetoma. Bonifaz et al.[9] treated a case of eumycetoma with a loading dose of 300 mg itraonazole, which was gradually tapered to 100 mg daily owing to an excellent response. Itraonazole has also been reported to be successful in treating mycetoma complicating other conditions with bone involvement.[10] We started our patient on oral itraconazole 200 mg daily monotherapy due to its good response, lower cost of therapy, and lower risk of serious adverse effects because the therapy requires a long duration. The patient showed a dramatic response and at 8 months of follow-up, all her lesions were cleared and she was completely cured. Her liver function tests at follow-ups had no alterations and there was no recurrence at 18 months of follow up. Resolution of pain and other depressive symptoms on antidepressant therapy point toward the importance of addressing psychological aspects of dermatological diseases.

Our patient responded very well to the therapy although most eumycetomas are considered to have a poor prognosis. We can conclude that mycetoma that does not involve deep structures such as bone can be treated medically with oral low-dose itraconazole, which is a cost-effective modality without fear of potential adverse effects.

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Conflicts of interest

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