Ferreira Mário, Paulo Joana, Ramos Paulo, Padrão Carolina, Neves Zélia
Serviço de Medicina Interna, Hospital Professor Doutor Fernando Fonseca, Lisboa, PRT.
Serviço de Anatomia Patológica, Hospital Professor Doutor Fernando Fonseca, Lisboa, PRT.
Cureus. 2024 Feb 2;16(2):e53468. doi: 10.7759/cureus.53468. eCollection 2024 Feb.
T-large granular lymphocytic leukemia (T-LGLL) is a rare lymphoproliferative disorder. The diagnosis is established by identifying an abnormally high number of clonal granular T lymphocytes in the peripheral blood and eventually in the bone marrow, in cases with medullary infiltration. The majority of patients present with symptoms related to neutropenia and this condition may be associated with autoimmune diseases in up to a third of cases. The authors describe the case of a 26-year-old patient admitted with subacute high fever and bullous dermatitis with necrotic lesions with central bullae. Analytically, she presented anemia and leukopenia with severe neutropenia of 200 cells/L. Skin lesions were compatible with ecthyma and the skin biopsy revealed aspects compatible with leukocytoclastic vasculitis. The myelogram and bone biopsy revealed hypoplasia of the myeloid line and a pathological T population of CD8+, TIA-1+ and granzyme B+, which were associated with compatible flow cytometry (CD3+, T-cell receptor (TCR) Alpha-Beta+, CD5+, CD2+, with loss of CD7 antigen expression) established the diagnosis of T-LGLL. The patient had a favorable evolution, with cytopenias almost returning to normal after two months. She began follow-up at a Hematology Reference Center, remaining asymptomatic without specific treatment considering the indolent course of the disease.
T 大颗粒淋巴细胞白血病(T-LGLL)是一种罕见的淋巴增殖性疾病。诊断通过在外周血中识别出异常大量的克隆性颗粒 T 淋巴细胞来确立,在骨髓浸润的病例中最终要在骨髓中识别。大多数患者表现出与中性粒细胞减少相关的症状,在多达三分之一的病例中,这种情况可能与自身免疫性疾病有关。作者描述了一名 26 岁患者的病例,该患者因亚急性高热和伴有中央大疱的坏死性病变的大疱性皮炎入院。分析显示,她出现贫血和白细胞减少,严重中性粒细胞减少至 200 个细胞/升。皮肤病变与深脓疱病相符,皮肤活检显示与白细胞破碎性血管炎相符的表现。骨髓检查和骨髓活检显示髓系发育不全以及 CD8 +、TIA-1 + 和颗粒酶 B + 的病理性 T 细胞群,这与相符的流式细胞术结果(CD3 +、T 细胞受体(TCR)α-β +、CD5 +、CD2 +,伴有 CD7 抗原表达缺失)共同确立了 T-LGLL 的诊断。患者病情进展良好,两个月后血细胞减少几乎恢复正常。她开始在血液学参考中心进行随访,考虑到疾病进展缓慢,在未进行特殊治疗的情况下一直无症状。