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Gamma 重链病:通过分析 13 例病例定义相关淋巴增殖性疾病的谱。

Gamma heavy-chain disease: defining the spectrum of associated lymphoproliferative disorders through analysis of 13 cases.

机构信息

Pathology and Laboratory Medicine Institute, Cleveland Clinic, Cleveland, OH 44195, USA.

出版信息

Am J Surg Pathol. 2012 Apr;36(4):534-43. doi: 10.1097/PAS.0b013e318240590a.

DOI:10.1097/PAS.0b013e318240590a
PMID:22301495
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC3715127/
Abstract

Gamma heavy-chain disease (gHCD) is defined as a lymphoplasmacytic neoplasm that produces an abnormally truncated immunoglobulin gamma heavy-chain protein that lacks associated light chains. There is scant information in the literature regarding the morphologic findings in this rare disorder, but cases have often been reported to resemble lymphoplasmacytic lymphoma (LPL). To clarify the spectrum of lymphoproliferative disorders that may be associated with gHCD, this study reports the clinical, morphologic, and phenotypic findings in 13 cases of gHCD involving lymph nodes (n=7), spleen (n=2), bone marrow (n=8), or other extranodal tissue biopsies (n=3). Clinically, patients showed a female predominance (85%) with frequent occurrence of autoimmune disease (69%). Histologically, 8 cases (61%) contained a morphologically similar neoplasm of small lymphocytes, plasmacytoid lymphocytes, and plasma cells that was difficult to classify with certainty, whereas the remaining 5 cases (39%) showed the typical features of one of several other well-defined entities in the 2008 WHO classification. This report demonstrates that gHCD is associated with a variety of underlying lymphoproliferative disorders but most often shows features that overlap with cases previously reported as "vaguely nodular, polymorphous" LPL. These findings also provide practical guidance for the routine evaluation of small B-cell neoplasms with plasmacytic differentiation that could represent a heavy-chain disease and give suggestions for an improved approach to the WHO classification of gHCD.

摘要

γ重链病(gHCD)定义为一种淋巴浆细胞肿瘤,其产生异常截断的免疫球蛋白γ重链蛋白,缺乏相关的轻链。关于这种罕见疾病的形态学发现,文献中仅有少量信息,但此类病例常被报道为类似于淋巴浆细胞淋巴瘤(LPL)。为了阐明可能与 gHCD 相关的淋巴增生性疾病的范围,本研究报告了 13 例涉及淋巴结(n=7)、脾脏(n=2)、骨髓(n=8)或其他结外组织活检(n=3)的 gHCD 的临床、形态和表型发现。临床上,患者表现为女性优势(85%),常伴有自身免疫性疾病(69%)。组织学上,8 例(61%)包含形态上相似的小淋巴细胞、浆细胞样淋巴细胞和浆细胞瘤的肿瘤,难以明确分类,而其余 5 例(39%)显示 2008 年 WHO 分类中几种其他明确实体的典型特征。本报告表明,gHCD 与多种潜在的淋巴增生性疾病相关,但最常见的表现与以前报道的“模糊结节状、多形性”LPL 病例重叠。这些发现还为常规评估具有浆细胞分化的小 B 细胞肿瘤提供了实用指导,这些肿瘤可能代表重链疾病,并为改进 gHCD 的 WHO 分类提供了建议。

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本文引用的文献

1
Marginal zone lymphomas with plasmacytic differentiation and related disorders.伴有浆细胞分化的边缘区淋巴瘤及相关疾病。
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Lymphoplasmacytic lymphoma and other non-marginal zone lymphomas with plasmacytic differentiation.淋巴浆细胞淋巴瘤和其他伴有浆细胞分化的非边缘区淋巴瘤。
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Monoclonal free light chains can be found in heavy chain diseases.单克隆游离轻链可在重链病中发现。
Ann Clin Biochem. 2010 Nov;47(Pt 6):570-2. doi: 10.1258/acb.2010.010146. Epub 2010 Oct 7.
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Fluorescence immunophenotypic and interphase cytogenetic characterization of nodal lymphoplasmacytic lymphoma.淋巴结浆细胞样淋巴瘤的荧光免疫表型及间期细胞遗传学特征
Am J Surg Pathol. 2008 Nov;32(11):1643-53. doi: 10.1097/PAS.0b013e3181758806.
6
Case records of the Massachusetts General Hospital. Case 13-2008. A 46-year-old man with rheumatoid arthritis and lymphadenopathy.马萨诸塞州总医院病例记录。病例13 - 2008。一名患有类风湿性关节炎和淋巴结病的46岁男性。
N Engl J Med. 2008 Apr 24;358(17):1838-48. doi: 10.1056/NEJMcpc0800959.
7
Gamma heavy chain disease in a patient with diabetes and chronic renal insufficiency: diagnostic assessment of the heavy chain fragment.一名患有糖尿病和慢性肾功能不全患者的γ重链病:重链片段的诊断评估
J Clin Lab Anal. 2008;22(2):146-50. doi: 10.1002/jcla.20233.
8
Extranodal marginal zone B-cell lymphomas of the ocular adnexa: multiparameter analysis of 34 cases including interphase molecular cytogenetics and PCR for Chlamydia psittaci.眼附属器结外边缘区B细胞淋巴瘤:34例的多参数分析,包括间期分子细胞遗传学和鹦鹉热衣原体的PCR检测
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9
Significantly improved PCR-based clonality testing in B-cell malignancies by use of multiple immunoglobulin gene targets. Report of the BIOMED-2 Concerted Action BHM4-CT98-3936.通过使用多个免疫球蛋白基因靶点,显著改进了B细胞恶性肿瘤中基于聚合酶链反应的克隆性检测。BIOMED-2协调行动BHM4-CT98-3936报告。
Leukemia. 2007 Feb;21(2):207-14. doi: 10.1038/sj.leu.2404479. Epub 2006 Dec 14.
10
t(14;18)(q32;q21) involving IGH and MALT1 is uncommon in cutaneous MALT lymphomas and primary cutaneous diffuse large B-cell lymphomas.涉及IGH和MALT1的t(14;18)(q32;q21)在皮肤黏膜相关淋巴组织淋巴瘤和原发性皮肤弥漫性大B细胞淋巴瘤中并不常见。
J Cutan Pathol. 2006 Apr;33(4):286-92. doi: 10.1111/j.0303-6987.2006.00459.x.