Temozolomide showed rapid activity in a patient with refractory CNS monomorphic epitheliotropic T cell lymphoma
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LETTER TO THE EDITOR
Temozolomide showed rapid activity in a patient with refractory CNS monomorphic epitheliotropic T cell lymphoma Pui-Lun Yip 1
&
Ching-Pong Lam 1 & Wai-Pan Lau 1 & Tsan-Hei Luk 1 & Sze-Man June Lau 1
Received: 6 September 2020 / Accepted: 21 September 2020 # Springer-Verlag GmbH Germany, part of Springer Nature 2020
Dear Editor, Monomorphic epitheliotropic intestinal T cell lymphoma (MEITL), previously known as enteropathy-associated T cell lymphoma (EATL) type II, is a rare T cell lymphoma. Prognosis is poor with a median survival of 7 months only [1]. Central nervous system (CNS) of MEITL was uncommonly reported, and there is no standard treatment. This prompts us to report our patient with high-dose methrotrexate (HD-MTX) refractory CNS MEITL, who responded effectively to temozolomide. A 67-year-old lady with history of diabetes mellitus presented with abdominal pain, weight loss and fever. A 14-cm pelvic mass was demonstrated on computed tomography (CT) of the abdomen and pelvis. Exploratory laparotomy revealed a perforated 14-cm ileal tumor and multiple enlarged mesenteric lymph nodes. Histopathological examination of the specimen diagnosed MEITL with diffuse mucosal infiltration by medium-sized monomorphic lymphocytes showing epitheliotropism. The lymphocytes were positive for CD3, CD8, CD56, and TIA. They were negative for CD20, CD5, and EBER-ISH. Six cycles of CHOP resulted in complete remission as shown on CT neck to pelvis. She complained of lower limb weakness and unsteady gait 1 month after the completion of CHOP. CT scan of the brain revealed a heterogeneous isodense mass with mass effect on left basal ganglia (Fig. 1a). The mass showed irregular contrast enhancement on magnetic resonance imaging (MRI) (Fig. 1d). Her neurological function further deteriorated—developing into right hemiparesis and aphasia then bed-ridden status despite the use of dexamethasone. Stereotactic biopsy of the brain lesion confirmed CNS involvement of MEITL. Two cycles of HD-
* Pui-Lun Yip [email protected] 1
Department of Medicine, Queen Elizabeth Hospital, 30 Gascoigne Road, Kowloon, Hong KongSAR
MTX were given, and high-dose cytarabine was added in the second cycle. CT scan of the brain showed the mass progressed (Fig. 1b). Temozolomide (150 mg/m2, 5 days in 28-day cycle) was initiated as salvage treatment. CT scan of the brain showed a significant reduction of the left basal ganglia mass after the first temozolomide cycle and the size further reduced after the second cycle (Fig. 1c). MRI of the brain after cycle 2 showed a largely resolved left basal ganglia tumor (Fig. 1e). MEITL is an exceedingly rare disease, and reports of CNS involvement in MEITL are scanty [2, 3]. Unlike in classical EATL which is often a small-bowel localized lymphoma, extra-intestinal involvement is frequently found in relapsed MEITL. In a case series by Chan et al. [3], parenchymal CNS involvement was reported in two out of the five relapsed or progressed MEITL. In another recent MEITL cohort in which more than 70% of t
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