Xu Victoria Y Y, Bell Mary, Zahirieh Alireza, Hsiao Janey, Higgins Kevin, Ghorab Zeina, Bookman Arthur, Chan Pak Cheung
Department of Medicine, University of Toronto, Toronto, ON, Canada.
Division of Rheumatology, Sunnybrook Health Sciences Centre, Toronto, ON, Canada.
Case Rep Med. 2017;2017:8748696. doi: 10.1155/2017/8748696. Epub 2017 Jul 30.
A 76-year-old man was incidentally found on a CT scan to have lymphadenopathy and bilateral kidney enlargement suggestive of infiltrative renal disease. He was largely asymptomatic but had bilateral salivary and lacrimal gland enlargement. A grossly elevated serum IgG (>70 g/L) with concomitant suppression of other immunoglobulins, a small IgG restriction, and a parotid biopsy revealing lymphoplasmacytic infiltrate with slight kappa light chain excess all suggested a lymphoproliferative disorder (LPD). The diagnostic workup was further confounded by a normal serum IgG4 concentration. Moreover, bone marrow and renal biopsies did not reveal evidence of LPD. Discussion with the laboratory not only clarified that the markedly increased total IgG could not be accounted for by the small IgG restriction, but also identified a discrepancy in the IgG4 measurement. Repeat analysis of a follow-up sample revealed an elevated IgG4 of 5.94 (reference interval: 0.039-0.864) g/L, which prompted a repeat parotid biopsy that showed predominant IgG4+ lymphocytic infiltrates. Despite the deluding presentations, a final diagnosis of IgG4-related disease (IgG4-RD) was made based on elevated serum IgG4 concentrations and histopathological findings. This case highlights the importance of recognizing limitations of laboratory testing and the benefit of close communications among clinical subspecialties and the laboratory.
一名76岁男性在CT扫描时偶然发现有淋巴结病和双侧肾脏肿大,提示存在浸润性肾病。他基本无症状,但有双侧唾液腺和泪腺肿大。血清IgG显著升高(>70 g/L),同时其他免疫球蛋白受到抑制,有轻微的IgG限制,腮腺活检显示淋巴细胞和浆细胞浸润,kappa轻链略有过量,所有这些都提示存在淋巴增殖性疾病(LPD)。血清IgG4浓度正常使诊断检查更加复杂。此外,骨髓和肾脏活检未发现LPD的证据。与实验室讨论不仅明确了IgG的轻微限制无法解释总IgG的显著增加,还发现了IgG4测量存在差异。对随访样本的重复分析显示IgG4升高至5.94(参考区间:0.039 - 0.864)g/L,这促使再次进行腮腺活检,结果显示主要为IgG4 + 淋巴细胞浸润。尽管有误导性表现,但基于血清IgG4浓度升高和组织病理学结果,最终诊断为IgG4相关疾病(IgG4 - RD)。该病例突出了认识实验室检测局限性的重要性以及临床亚专业与实验室之间密切沟通的益处。