Ulcerative Colitis Concomitant with Cytomegalovirus Infection, Bullous Sweet's Syndrome, and Acute Myeloid Leukemia: A Case Report and Literature Review

Abstract

Background: Ulcerative colitis (UC) is a chronic, relapsing progressive inflammatory immune disease. There is still no cure for it. Even worse, UC may predispose patients to opportunistic infections, and several extra-intestinal manifestations (EIMs) and comorbidities may antedate, occur with, or postdate the onset of UC, which may increase the mortality risk. But case reports of UC patients simultaneously concomitant with opportunistic infection, EIM, and comorbidity are extremely rare.

Case presentation: We report a case of 51-year-old male patient with incipient UC accompanied by cytomegalovirus (CMV) infection and bullous Sweet's syndrome (bSS, a cutaneous EIM of UC) after treatment with oral mesalazine and prednisolone for 3 weeks. After clearance of the CMV infection by using ganciclovir, the patient was administered two cycles of infliximab to cure UC and bSS; however, he developed acute myeloid leukemia (AML) a month later and died after two cycles of chemotherapy.

Conclusion: Based on this rare case of UC concomitant with CMV infection, bSS and AML, we recommend that it is important to distinguish between an acute UC flare and opportunistic infections, especially in patients receiving immunosuppressive therapy, and monitor EIMs and comorbidities timely. Particular attention should be paid to cancer surveillance. Clinicians should be mindful of these facts to adopt optimal therapeutic options to address all aspects of UC. Early initiation of biological therapy may be of benefit to patients with newly diagnosed severe UC.

Keywords: acute myeloid leukemia; bullous Sweet’s syndrome; cytomegalovirus; ulcerative colitis.

Figure 1

Total colonoscopy images. (A–D) First procedure performed on January 9, 2022, showing mucosal redness, edema, granular change, and multiple small mucosal ulceration. (E–H) Second procedure performed on January 28, 2022, showing multiple deep oval ulcers (arrows).

Figure 2

Abdominal CT scan findings. Image showing diffuse thickening of the colonic wall of ascending and descending colon (A) and sigmoid colon (B) (arrows), likely secondary to inflammation.

Figure 3

Photographs showing skin lesions on the patient’s arms, neck, and chest. (A) Initial Purple-erythematous infiltrating skin plaques on a photograph obtained on February 3. (B and C) Photograph obtained on February 6 (B) and on February 8 (C) showing progression of the skin lesions, presenting with bullous, papulovesicular, and pustular skin lesions.

Figure 4

Photographs showing clinical remission of skin lesions after administration of IFX. (A) Skin lesions observed after the first cycle of IFX treatment. (B) Lesions observed before administration of the second cycle and, (C) Before administration of the third cycle of IFX therapy.