Zhang Ranran, Liu Jia, Nie Nana, Wang Dahai, Wu Jie, Zhang Huanyu, Zhang Ruiyun, Gao Shan, Bai Cui, Lin Yi, Zhang Qiuye, Chang Hong
Department of Pediatric Nephrology, Rheumatology and Immunity, The Affiliated Hospital of Qingdao University, Qingdao, China.
Department of Pathology, The Affiliated Hospital of Qingdao University, Qingdao, China.
Front Pediatr. 2024 Nov 22;12:1469495. doi: 10.3389/fped.2024.1469495. eCollection 2024.
Castleman disease (CD) is a rare lymphoproliferative disease known as angiofollicular lymph node hyperplasia, firstly reported in 1954. It mainly occurs in adults, presenting with a wide range of clinical manifestations, including paraneoplastic pemphigus (PNP). PNP is a rare and often life-threatening autoimmune disorder characterized by painful blisters and erosions on the skin and mucous membranes. In children, PNP is often linked to Castleman disease, as evidenced in case reports. So far, less than 30 pediatric cases have been reported, with the pathogenesis remaining unclear and treatment approaches varied. Here, we present a pediatric case initially suspected as Behçet's disease due to persistent oral ulcers and conjunctivitis, and undergone a sudden aggravation of clinical features following an allergic reaction. New involvement of skin rashes and imaging findings prompted the final diagnosis as PNP linked to Unicentral Castleman disease (UCD).Through detailing the progression of clinical features and diagnostic work, we aim to arise the awareness of physicians and put emphasize on early recognition and multidisciplinary management, which can improve patient outcomes.
卡斯特曼病(CD)是一种罕见的淋巴增生性疾病,被称为血管滤泡性淋巴结增生,于1954年首次报道。它主要发生于成年人,临床表现多样,包括副肿瘤性天疱疮(PNP)。PNP是一种罕见且常危及生命的自身免疫性疾病,其特征为皮肤和黏膜出现疼痛性水疱和糜烂。在儿童中,PNP常与卡斯特曼病相关,病例报告已有证明。迄今为止,报道的儿科病例少于30例,发病机制尚不清楚,治疗方法也各不相同。在此,我们报告一例儿科病例,该患儿最初因持续性口腔溃疡和结膜炎被怀疑为白塞病,在发生过敏反应后临床特征突然加重。新发皮疹及影像学检查结果促使最终诊断为与单中心卡斯特曼病(UCD)相关的PNP。通过详细阐述临床特征的进展及诊断过程,我们旨在提高医生的认识,并强调早期识别和多学科管理,以改善患者预后。