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累及唾液腺的抗 CD19 CAR-T 治疗后器官特异性局部细胞因子释放综合征:一例病例报告、文献综述及对托珠单抗相关颈部肿胀的诊断警示

英文原题:Organ-specific local cytokine release syndrome after anti-CD19 CAR-T therapy with salivary gland involvement: a case report, literature review, and a diagnostic alert for tocilizumab-associated cervical swelling.

查看英文原题

Organ-specific local cytokine release syndrome after anti-CD19 CAR-T therapy with salivary gland involvement: a case report, literature review, and a diagnostic alert for tocilizumab-associated cervical swelling.

PubMed 2026/04/22(内容时间) Ann Hematol Q3 · IF 2.3(JCR 2025)

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中文摘要

背景。细胞因子释放综合征(CRS)是CAR-T 细胞治疗后最常见的毒性反应,通常表现为全身性炎症综合征。近年来,一种局限性CRS(L-CRS)越来越多地被认识,最常累及颅颈区域。L-CRS可伴有或不伴有明显的局部肿瘤受累,并可进展为气道损害,需要及时识别和治疗。病例介绍。

我们报告两例接受抗CD19 CAR-T 治疗的难治性弥漫性大B细胞淋巴瘤(DLBCL)患者,在输注后早期、全身性CRS经tocilizumab治疗之后,突然出现疼痛性双侧腮腺和/或颌下腺肿胀(以左侧为主)。感染性、梗阻性和自身免疫性病因均已排除;超声表现符合炎性腺体改变。鉴于病情快速进展及对气道通畅的担忧,给予糖皮质激素治疗,临床症状迅速缓解。文献回顾与诊断警示。

我们概述了已发表的表现为颅颈症状的L-CRS(表1)。在其他情况下也曾报道与tocilizumab时间相关的类似急性肿胀。

在此类病例中,需要对L-CRS与tocilizumab相关输注反应/超敏反应进行鉴别诊断。结论。L-CRS是CAR-T 治疗的一种具有临床意义且可能严重的并发症,即使没有颈部肿瘤负荷也可能累及唾液腺。临床医生还应考虑药物相关反应,尤其是当颈部肿胀发生在tocilizumab给药后不久时。

展开英文摘要原文

Background. Cytokine release syndrome (CRS) is the most frequent toxicity after chimeric antigen receptor T-cell (CAR-T) therapy and typically presents as a systemic inflammatory syndrome. In recent years, a localized form of CRS (L-CRS), most commonly involving the craniocervical region, has been increasingly recognized. L-CRS may occur with or without overt local tumor involvement and can progress to airway impairment, requiring prompt recognition and treatment. Case presentation.

We report two cases of patients with refractory diffuse large B-cell lymphoma (DLBCL) treated with anti-CD19 CAR-T therapy who developed abrupt, painful bilateral parotid and/or submandibular gland swelling (left predominance) early after infusion, following systemic CRS treated with tocilizumab.

Infectious, obstructive, and autoimmune causes were excluded; ultrasound findings were compatible with inflammatory glandular changes. Given rapid progression and concern for airway patency, corticosteroids were administered with prompt clinical resolution. Literature review and diagnostic alert.

We provide an overview of published L-CRS with craniocervical presentations (Table 1). Similar acute swelling temporally related to tocilizumab has been reported in other settings. In such cases, differential diagnostics of L-CRS vs. a tocilizumab-associated infusion-related/hypersensitivity reaction needs to be carried out.

Conclusion. L-CRS is a clinically relevant and potentially severe complication of CAR-T therapy that may involve salivary glands even without cervical tumor burden. Clinicians should also consider drug-related reactions, particularly when cervical swelling occurs shortly after tocilizumab administration.

论文信息

作者
Maggi R、Ricciuti EF、Galli E、Sorà F、Fiorita A、Sica S、Chiusolo P
单位
Hematology Division, Policlinico Universitario Agostino Gemelli, Largo Francesco Vito 1, Rome, Italy. robertomaggi20@gmail.com.France
文献类型
病例报告 · 综述
期刊
Annals of hematology2026 Apr 22
原文标识
PubMed 42014477 · DOI 10.1007/s00277-026-07013-0