部分质体树突状细胞分化的高级别髓系恶性肿瘤的诊断难题:两例病例报告及文献综述。

IF 2.2 4区 医学 Q3 HEMATOLOGY Leukemia & Lymphoma Pub Date : 2024-12-20 DOI:10.1080/10428194.2024.2422846
Karen Amelia Nahmod, Roberto N Miranda, Francisco Vega, Beenu Thakral, Naveen Pemmaraju, Guillermo Montalban-Bravo, Sanam Loghavi, Fatima Zahra Jelloul, Wei Wang, Sa Wang, Tariq Muzzafar, Keyur Patel, Carlos E Bueso-Ramos, L Jeffrey Medeiros, Rashmi Kanagal-Shamanna
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引用次数: 0

摘要

髓系肿瘤伴浆细胞样树突状细胞(pDC)分化的诊断具有挑战性,因为免疫表型重叠,需要流式细胞术和免疫组织化学进行详细的表征。我们描述了两例有骨髓增生性肿瘤(MPN)和骨髓增生异常/骨髓增生性肿瘤(MDS/MPN)病史的患者,他们在数年后出现白细胞增多、淋巴结病、脾肿大和恶病质,临床迅速恶化并死亡。淋巴结活检标本显示髓样肉瘤累及,有明显的pDC分化。此外,同时进行的骨髓穿刺和活检显示潜在的髓系肿瘤浸润,但在淋巴结标本中未见pDC平行扩张,这表明pDC分化是在淋巴结微环境中培养的。这两个病例可能代表了最近描述的急性髓系白血病伴pDC分化(pDC- aml)的“髓系肉瘤”。尽管pDC- aml患者在接受常规治疗时预后较差,但在这些肿瘤中发现pDC成分可能会扩大治疗选择。
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Diagnostic challenges of high-grade myeloid malignancies with partial plasmacytoid dendritic cell differentiation: report of two cases with literature review.

The diagnosis of myeloid neoplasms with plasmacytoid dendritic cell (pDC) differentiation can be challenging due to immunophenotypic overlap requiring detailed characterization by flow cytometry and immunohistochemistry. We describe two patients with a history of myeloproliferative neoplasm (MPN) and myelodysplastic/myeloproliferative neoplasm (MDS/MPN) who presented years later with leukocytosis, lymphadenopathy, splenomegaly, and cachexia, with rapid clinical deterioration and death. Lymph node biopsy specimens revealed involvement by myeloid sarcoma with prominent pDC differentiation. Furthermore, concomitant bone marrow aspiration and biopsy showed involvement by the underlying myeloid neoplasm but no parallel expansion of pDC, as seen in the lymph node specimens, suggesting that pDC differentiation is fostered in the lymph node microenvironment. These two cases could represent the "myeloid sarcoma" counterpart of the recently described acute myeloid leukemia with pDC differentiation (pDC-AML). Although patients with pDC-AML have an inferior outcome when treated with conventional therapies, the recognition of a pDC component in these neoplasms potentially expands the therapeutic options.

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来源期刊
Leukemia & Lymphoma
Leukemia & Lymphoma 医学-血液学
CiteScore
4.10
自引率
3.80%
发文量
384
审稿时长
1.8 months
期刊介绍: Leukemia & Lymphoma in its fourth decade continues to provide an international forum for publication of high quality clinical, translational, and basic science research, and original observations relating to all aspects of hematological malignancies. The scope ranges from clinical and clinico-pathological investigations to fundamental research in disease biology, mechanisms of action of novel agents, development of combination chemotherapy, pharmacology and pharmacogenomics as well as ethics and epidemiology. Submissions of unique clinical observations or confirmatory studies are considered and published as Letters to the Editor
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