An Unusual Case of EHBO

Introduction: Chromoblastomycosis is a chronic fungal infection involving skin and subcutaneous tissue resulting from implantation of certain dematiaceous fungi secondary to minor puncture wounds from plants. It is an orphan neglected disease that usually prevail in tropical and subtropical countries. It starts as a macular lesion at inoculation site which slowly progresses to papulosquamous lesion. Extrahepatic biliary obstruction commonly occurs due to bile duct stones, neoplastic lesions, inflammatory condition; however, biliary obstruction due to fungal infection is rare and it is even rarer in immunocompetent patients.

Case Description: We reported a case of 29-year-old male patient presented with multiple papular skin lesions on left cheek and lower limbs since last 15 years which gradually progressed to large verrucous plaque-like lesions along with painless progressive cholestatic jaundice and generalized pruritus since 2 months with clay colored stools. History of low grade fever with significant anorexia and weight loss of 10–15 kg in the last 10–15 years. History of ATT intake for 1 month one year back due to suspicion of tubercular skin lesions was also present. On blood investigations liver functions test were deranged showing cholestatic pattern with total bilirubin 5.5 mg/dL with direct fraction 4.5 mg/dL, transaminitis (SGOT/SGPT 98/113 U/L) and SAP 1180 U/L, GGT 521 U/L.

USG W/A showed distended GB with grossly dilated CBD measuring 29 mm with bilobar IHBRD. MRCP also showed dilated IHBR with pruning of peripheral IHBR and diffuse irregular circumferential wall thickening with diffusion restriction involving CBD,CHD and central IHBR. . Biopsies from cervical lymph nodes, ampullary biopsy and skin punch biopsy from left cheek all showed presence of fungal hyphae and spores which were positive on PAS and GMS staining. Also on skin punch biopsy from cheek revealed pseudoepitheliomatous hyperplasia with intraepidermal abscess with pigmented sclerotic bodies (medlar and copper bodies) which is characteristic of a particular group of fungi named chromoblastomycosis. A diagnosis of extrahepatic biliary obstruction with disseminated chromoblastomycosis with secondary sclerosing cholangitis was made. He was managed with ERCP and DPT placement in CBD and started on antifungals (Tab Itraconazole 200 mg twice daily combined with Tab terbinafine 250 mg once daily). Follow-up after 2 months, the patient's appetite had improved with resolution of pruritus and lower limb skin lesions. Repeat biopsy from ampulla was negative for fungal hyphae or spores.

Discussion: Fungal infections involving hepatobiliary tree are rare. Pathogenesis postulated for biliary tract involvement by fungi are direct invasion of the common bile duct through duodenum hematogenous spread from underlying systemic fungal infection. In our patient, hematogenous route seems likely since the patient had underlying systemic manifestations. This patient had disseminated chromoblastomycosis with biliary obstruction, thus we report the first case of biliary obstruction due to chromoblastomycosis.

Publication History

Article published online:
22 April 2024