Division of Cardiothoracic Surgery, The Valley Hospital, 223 N Van Dien Ave, Ridgewood, NJ, 07450, USA.
Division of Radiology, The Valley Hospital, Ridgewood, NJ, 07450, USA.
J Cardiothorac Surg. 2024 Oct 8;19(1):599. doi: 10.1186/s13019-024-03026-w.
A 54-year-old female presented with recurrent abdominal pain and new onset chest pain. Chest computed-tomography angiogram detected a thoracic aortic aneurysm with suspected Type A intramural hematoma (IMH) versus aortitis. Initially, conservative management was pursued while awaiting a definitive diagnosis. Differential workup was negative, while additional imaging modalities favored IMH, prompting expedited surgical intervention. During ascending aortic and hemiarch replacement, severe aortitis was unexpectedly discovered without evidence of IMH. Histopathological examination of the aortic specimens identified transmural aortic inflammation with lymphoplasmacytic infiltrate and irregular fibrosis. Numerous IgG4-positive plasma cells were present with IgG4/IgG ratio of 40-50% suggesting IgG4-related disease (IgG4-RD). Subsequent analysis revealed B cells positive for clonal IgH gene rearrangement, and bone marrow biopsy then revealed the same clonal B cells. She was ultimately diagnosed with CLL, the most common phenotype of monoclonal B-cell lymphocytosis, thought to account for the IgG4-predominant plasma cells causing aortitis. Although rare, this case highlights the importance of considering IgG4-related disease (IgG4-RD) as a cause of aortitis when assessing symptomatic patients with aortic pathologies, emphasizing the complexities involved in diagnosing due to a variety of imaging presentation, differentiating, and managing large-vessel vasculitides. Moreover, it underscores the importance of Multidisciplinary Aortic Team care and the use of multiple diagnostic modalities in evaluating ambiguous aortic pathologies.
一位 54 岁女性因反复腹痛和新发胸痛就诊。胸部 CT 血管造影检查发现胸主动脉瘤,疑似 A 型壁内血肿(IMH)与主动脉炎。最初,在等待明确诊断的同时,采取了保守治疗。鉴别诊断检查结果为阴性,而其他影像学检查更倾向于 IMH,促使进行紧急手术干预。在升主动脉和半弓置换术中,意外发现严重的主动脉炎,而没有 IMH 的证据。主动脉标本的组织病理学检查显示,存在贯穿性主动脉炎症,伴有淋巴浆细胞浸润和不规则纤维化。有大量 IgG4 阳性浆细胞,IgG4/IgG 比值为 40-50%,提示 IgG4 相关疾病(IgG4-RD)。进一步分析显示 B 细胞存在克隆性 IgH 基因重排,骨髓活检也显示相同的克隆性 B 细胞。最终诊断为 CLL,这是单克隆 B 细胞淋巴细胞增多症最常见的表型,被认为是导致主动脉炎的 IgG4 占优势的浆细胞。尽管罕见,但该病例强调了在评估有主动脉病变症状的患者时,应考虑 IgG4 相关疾病(IgG4-RD)作为主动脉炎的病因的重要性,这凸显了由于各种影像学表现、鉴别和管理大血管血管炎的复杂性。此外,它强调了多学科主动脉团队治疗和在评估不明确的主动脉病变时使用多种诊断方法的重要性。