Pathology and Laboratory Medicine Institute, Cleveland Clinic, Cleveland, OH 44195, USA.
Am J Surg Pathol. 2012 Apr;36(4):534-43. doi: 10.1097/PAS.0b013e318240590a.
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.
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。