Maintenance and preemptive therapy with ganciclovir for cytomegalovirus colitis with extremely high antigenemia in adult T-cell leukemia

Abstract

Patients with adult T-cell leukemia (ATL) are susceptible to opportunistic infections. The major cause of death in ATL patients is cytomegalovirus (CMV) pneumonia [1]. However, the onset of CMV infection in a very early phase of ATL is uncommon [2]. In addition, there are no detailed descriptions concerning the effect of oral ganciclovir (GCV) in ATL. Herein, we report a case with ATL who demonstrated CMV colitis on the second day of prednisolone administration. A 48-year-old female was admitted because of fever, erythema, and fatigue. Her white blood cell (WBC) count was 18,400/mm (atypical lymphocytes including flower cells: 48.5%). The left and right cervical, axillary, and right inguinal lymph nodes were swollen. Erythema of the face, limbs, and neck was observed. Histopathologic findings of biopsy specimens from erythema sites demonstrated the possibility of ATL cells having invaded the skin. The lactate dehydrogenase (LDH) score was 505 U/L (more than twice the normal upper limit) and human T lymphotrophic virus type 1 provirus was detected on Southern blot analysis. Therefore, we diagnosed this case as acutetype ATL. The compensated calcium level was normal (9.6 mg/dL), and there were no other sites of involvement such as the liver, spleen, and cerebrospinal fluid. She was started on prednisolone for ATL. On the day following the start of administration, diarrhea and abdominal pain developed. We started chemotherapy primarily according to the LSG 15 protocol [3], because it was possible at that time that her diarrhea was caused by ATL cell invasion of the colon. However, an abdominal computed tomography scan showed ascites and mucosal thickening of the colon, which indicated colitis. Histopathologic findings of biopsy specimens from edematous lesions of the rectum detected by proctoscopy demonstrated intranuclear CMV inclusion bodies in endothelial cells but not the invasion of ATL cells. In addition, there were 3411 and 3258 CMV antigenpositive leukocytes each per 15 9 10 WBCs, and other pathogens which can induce diarrhea were not detected. No other manifestations induced by CMV infection such as CMV pneumonia and retinitis appeared. Therefore, the diagnosis of CMV colitis was confirmed. She received intravenous GCV. Diarrhea improved and the score of CMV antigenemia gradually declined (Fig. 1). After the diarrhea had fully resolved and informed consent was given, we continued maintenance therapy with oral GCV until CMV antigenemia became negative. Thereafter, we chose preemptive therapy involving oral GCV along with the monitoring of CMV antigenemia. This patient received allogeneic bone marrow transplantation (BMT) four months after the diagnosis of CMV colitis. We continued the same preemptive therapy until BMT. CMV infection never recurred before BMT. In addition, chemotherapy was effective. After chemotherapy, atypical lymphocytes in the peripheral blood disappeared, and the score of the soluble interleukin-2 receptor was reduced (Fig. 1). Swollen lymph nodes regressed. In addition, the level of LDH was reduced, but just less than half of the erythematous area S. Nagai M. Toshima K. Sato M. Mori T. Nagai K. Muroi K. Ozawa Division of Hematology, Department of Medicine, Jichi Medical University, 3311-1 Yakushiji, Shimotsuke, Tochigi 3290498, Japan