Abstract
Introduction:
Eosinophilic colitis (EC) is a rare inflammatory condition involving eosinophilic infiltration of the mucosa in the absence of a secondary cause or peripheral eosinophilia. Clinically, EC presents with non-specific symptoms. We present a case of malignant EC that is refractory to different therapies.
Case Description/Methods:
A 30 years old male with known EC on azathioprine, pulmonary embolism and superior mesenteric venous (SMV) thrombosis onrivaroxaban, had multiple admissions with abdominal pain and hematochezia secondary to Clostridioides difficile (C. diff). His rivaroxaban and azathioprine were held given his hematochezia and recurrent infections. He presented a month later with worsening symptoms but C. diff was negative. CT abdomen/pelvis showed non-occlusive SMV and portal venous (PV) thrombosis with signs of ischemia of the sigmoid colon (Figure 1b). Patient underwent percutaneous transhepatic SMV/PV aspiration thrombectomy that was complicated by a large right hemothorax, causing hemorrhagic shock. Subsequent flexible sigmoidoscopy showed signs of colonic ischemia (Figure 1a). Colonic biopsies demonstrated active colitis without evidence of chronicity, and 60 eosinophils per high powered field. Patient underwent a subtotal colectomy with end ileostomy with clinical improvement immediately postoperatively. He was discharged on a steroid taper and apixaban. He was started on the interleukin-5 (IL-5) inhibitor benralizumab for his EC. Outpatient follow-up was notable for continued rectal bleeding. Flexible sigmoidoscopy showed diffuse severe erythema, contact bleeding, and friable mucosa throughout the rectal pouch (Figure 1c). Vedolizumab was started in combination with benralizumab, however, on long-term outpatient follow-up, no notable clinical or endoscopic improvements were noted on combination therapy. Multi-disciplinary decision in conjunction with patient resulted in planned proctocolectomy with permanent end ileostomy.
Discussion:
There are currently no FDA approved management options for EC. The IL-5 inhibitor Benralizumab is FDA approved for treatment of eosinophilic asthma and therefore it was worth a trial in this patient. In a small number of case reports, Vedolizumab has shown some clinical improvement as well as reduction in eosinophilia in EC patients. Due to the rarity of EC diagnosis, data for therapies in EC is severely lacking. To our knowledge, this is the first case of a patient with severe refractory EC that led to the development of ischemic colitis.
Eosinophilic colitis (EC) is a rare inflammatory condition involving eosinophilic infiltration of the mucosa in the absence of a secondary cause or peripheral eosinophilia. Clinically, EC presents with non-specific symptoms. We present a case of malignant EC that is refractory to different therapies.
Case Description/Methods:
A 30 years old male with known EC on azathioprine, pulmonary embolism and superior mesenteric venous (SMV) thrombosis onrivaroxaban, had multiple admissions with abdominal pain and hematochezia secondary to Clostridioides difficile (C. diff). His rivaroxaban and azathioprine were held given his hematochezia and recurrent infections. He presented a month later with worsening symptoms but C. diff was negative. CT abdomen/pelvis showed non-occlusive SMV and portal venous (PV) thrombosis with signs of ischemia of the sigmoid colon (Figure 1b). Patient underwent percutaneous transhepatic SMV/PV aspiration thrombectomy that was complicated by a large right hemothorax, causing hemorrhagic shock. Subsequent flexible sigmoidoscopy showed signs of colonic ischemia (Figure 1a). Colonic biopsies demonstrated active colitis without evidence of chronicity, and 60 eosinophils per high powered field. Patient underwent a subtotal colectomy with end ileostomy with clinical improvement immediately postoperatively. He was discharged on a steroid taper and apixaban. He was started on the interleukin-5 (IL-5) inhibitor benralizumab for his EC. Outpatient follow-up was notable for continued rectal bleeding. Flexible sigmoidoscopy showed diffuse severe erythema, contact bleeding, and friable mucosa throughout the rectal pouch (Figure 1c). Vedolizumab was started in combination with benralizumab, however, on long-term outpatient follow-up, no notable clinical or endoscopic improvements were noted on combination therapy. Multi-disciplinary decision in conjunction with patient resulted in planned proctocolectomy with permanent end ileostomy.
Discussion:
There are currently no FDA approved management options for EC. The IL-5 inhibitor Benralizumab is FDA approved for treatment of eosinophilic asthma and therefore it was worth a trial in this patient. In a small number of case reports, Vedolizumab has shown some clinical improvement as well as reduction in eosinophilia in EC patients. Due to the rarity of EC diagnosis, data for therapies in EC is severely lacking. To our knowledge, this is the first case of a patient with severe refractory EC that led to the development of ischemic colitis.
Original language | American English |
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Journal | American Journal of Gastroenterology |
State | Published - Oct 2022 |