A 35 years-old woman from Kerman province has come to our clinic with abdominal pain mainly in the hypogastric area, constipation, and hematochezia. colonoscopy showed multiple erosions and ulcers in the sigmoid and descending colon with skip area. Based on colonoscopy and histopathology she had been diagnosed with Crohn's disease and mesalamine was prescribed for her. After a while due to incomplete symptom response. Her physician initiated Adalimumab. After initiation of Adalimumab, she noticed abdominal distension. At this time, she was referred to a tertiary center for more evaluation. With the impression of refractory Crohn's disease infliximab (Remicade) was started. Starting remicade was followed by increasing abdominal distension and perianal induration. MRI revealed sinusitis and extensive inflammation and swelling in pelvic floor muscles leading to anal canal narrowing. It also showed pan colitis and terminal ileitis with an active pattern in the rectosigmoid (Fig. 3).
The patient underwent a surgical biopsy of perianal tissue. microscopic examination showed poorly formed granuloma with central necrosis and occasional PMN aggregates and prominent infiltration of eosinophils. Ziehl-Neelsen and PAS staining were negative for acid-fast bacilli and fungal elements respectively. According to the finding of granulomatosis reaction and endemicity of TB in IRAN and the fact that symptoms worsened after anti-TNF therapy, anti-TB treatment was started at that center. She was referred to our hospital at this stage which she developed acute drug-induced hepatitis.
Despite anti-TB medications, there was still leukocytosis and elevated ESR and CRP. Our DDxs at this time were: pelvic Wegner disease, Histiocytosis X, lymphoma, Crohn's disease with cutaneous involvement and rare causes of gastrointestinal granulomatosis such as syphilis, Bartonella henselae, Basidiobolomycosis, and salmonellosis. Although we had negative results for fungal infection we started empirical amphotericin. No improvement was reached after two weeks. cystoscopy showed severe inflammation and erythema with severe PMN infiltration. Bone marrow biopsy was negative for infiltrative disease such as lymphoma. Laparoscopic exploration revealed distended bowel loops but no obvious mass. The laboratory results are in Table 1.
We reviewed all of the patient's histopathology samples to come up with a diagnosis with another expert pathologist. The pathologist diagnosed basidiobolomycosis by viewing the Splendore Hoeppli phenomenon and fungal element in GMS staining in samples of perianal biopsy (Fig. 4).
Itraconazole and co-trimoxazole were started after consultation with infectious disease specialists who had similar cases. One week later the patient's symptoms and laboratory indices (ESR, CRP, anemia) improved significantly (Table 1). Perineal induration was also resolved. The patient was discharged three weeks later. Itraconazole and co-trimoxazole were continued for 9 months. We were curious about the source of the infection and after asking for possible exposures we found out that the patient had been culturing sesame in her house for her Ph.D. thesis (Fig. 5). The only problem at the time of discharge was abdominal distention which had not been fully resolved. Three and twelve-month later at the follow-up CT scan there were no obstructions and the distension was attributed to the dysmotility of the sigmoid part of the colon which is a sequela of sigmoid involvement by basidiobolomycosis (Fig. 6). Now one year later after discharging the patient at the follow-up visits, she is in good condition.