Saparov Dosbai, Gold-Olufadi Shakirat, Wasifusddin Mustafa, Hakobyan Narek, Uche Ifeanyi, Becerra Henry, Barakat Philipp, Yadav Ruchi, Pokhrel Akriti, Boris Avezbakiyev, Wang Jen
Internal Medicine, Brookdale University Hospital Medical Center, Brooklyn, USA.
Medicine, Brookdale University Hospital Medical Center, Brooklyn, USA.
Cureus. 2024 Sep 27;16(9):e70325. doi: 10.7759/cureus.70325. eCollection 2024 Sep.
Castleman disease (CD) is a rare lymphoproliferative disorder with unicentric (UCD) and multicentric (MCD) forms, first detailed by Benjamin Castleman in 1956. It has three subtypes: hyaline vascular, plasma cell, and mixed. CD incidence is higher in HIV patients and is often associated with human herpes virus-8 (HHV-8). We report a 68-year-old woman with diabetes and mixed connective tissue disease (MCTD), which was diagnosed six months prior to presentation, who presented with lymphadenopathy, splenomegaly, and B symptoms. Imaging showed diffuse adenopathy. Biopsy confirmed the plasma cell subtype of MCD, with polyclonal plasmacytosis. The patient tested negative for HIV and HHV-8. Initial treatment with rituximab and corticosteroids resolved her symptoms. Six years later, she relapsed and was treated with an anti-IL-6 agent, which she could not complete due to adverse effects but still showed symptom improvement. This case is notable for the patient's age and the plasma cell subtype of MCD, as well as the concurrent diagnosis of MCTD. The patient's clinical presentation and histopathological findings underscore the importance of considering CD in the differential diagnosis of lymphadenopathy with systemic symptoms. Chronic inflammation and B lymphocyte proliferation, potentially linking MCTD and CD, were evident in this case. Despite extensive comorbidities, the patient remained clinically stable due to intensive multidisciplinary management. CD is a rare, heterogeneous disorder requiring a high index of suspicion. The potential link between CD and MCTD warrants further research. Effective management involves targeted therapies and close monitoring due to relapse risk. This case underscores the importance of individualized treatment plans considering comorbidities and treatment tolerability. Further research is needed to better understand CD's pathogenesis and develop effective treatments.
卡斯尔曼病(CD)是一种罕见的淋巴增生性疾病,有单中心型(UCD)和多中心型(MCD)两种形式,1956年由本杰明·卡斯尔曼首次详细描述。它有三种亚型:透明血管型、浆细胞型和混合型。CD在艾滋病患者中的发病率较高,且常与人类疱疹病毒8型(HHV-8)相关。我们报告一名68岁患有糖尿病和混合性结缔组织病(MCTD)的女性,MCTD在就诊前6个月被诊断出来,她出现了淋巴结病、脾肿大和B症状。影像学检查显示弥漫性淋巴结病。活检证实为MCD的浆细胞亚型,伴有多克隆浆细胞增多。该患者HIV和HHV-8检测均为阴性。最初使用利妥昔单抗和皮质类固醇治疗使她的症状得到缓解。6年后,她复发,接受了抗IL-6药物治疗,但由于不良反应未能完成治疗,但症状仍有改善。该病例值得注意的是患者的年龄、MCD的浆细胞亚型以及同时诊断的MCTD。患者的临床表现和组织病理学发现强调了在伴有全身症状的淋巴结病鉴别诊断中考虑CD的重要性。在该病例中,慢性炎症和B淋巴细胞增殖明显,这可能将MCTD和CD联系起来。尽管存在广泛的合并症,但由于多学科的强化管理,患者临床保持稳定。CD是一种罕见的异质性疾病,需要高度怀疑。CD与MCTD之间的潜在联系值得进一步研究。由于存在复发风险,有效的管理包括靶向治疗和密切监测。该病例强调了考虑合并症和治疗耐受性的个体化治疗方案的重要性。需要进一步研究以更好地理解CD的发病机制并开发有效的治疗方法。