儿童期系统性红斑狼疮(cSLE)与恶性肿瘤:全国多中心系列回顾。

IF 4.6 Q2 MATERIALS SCIENCE, BIOMATERIALS ACS Applied Bio Materials Pub Date : 2024-02-06 DOI:10.1186/s42358-024-00353-3
Matheus Zanata Brufatto, Sean Hideo Shirata Lanças, Taciana de Albuquerque Pedrosa Fernandes, Adriana Maluf Elias Sallum, Lucia Maria Arruda Campos, Ana Paula Sakamoto, Maria Teresa Terreri, Flavio Roberto Sztajnbok, Blanca Elena Rios Gomes Bica, Virginia Paes Leme Ferriani, Luciana Martins de Carvalho, Clovis Artur Almeida Silva, Claudia Saad-Magalhaes
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引用次数: 0

摘要

背景:成人系统性红斑狼疮(SLE)中恶性肿瘤发生率增高的情况已得到充分证实,但在儿童期发病的系统性红斑狼疮(cSLE)系列中的报道却很有限。我们探讨了儿童系统性红斑狼疮伴发恶性肿瘤的频率,描述了恶性肿瘤确诊时的临床和人口学特征、疾病活动和累积损害:方法:在27个儿科风湿病学中心的全国性队列中进行回顾性病例记录审查,每个参与中心均采用标准方案对活检证实的恶性肿瘤、疾病活动/损害累积和免疫抑制治疗进行了描述性汇编:结果:在更新队列的1757例系统性红斑狼疮病例中,有12例(0.7%)发生了恶性肿瘤,中位时间为确诊系统性红斑狼疮后10年。其中91%为女性,确诊系统性红斑狼疮的中位年龄为12岁,确诊恶性肿瘤的中位年龄为23岁。在所有确诊的恶性肿瘤中,11例为单部位恶性肿瘤,1例同时伴有多部位恶性肿瘤;4例为血液恶性肿瘤(0.22%),8例为实体恶性肿瘤(0.45%)。SLEDAI-2 K评分的中位数(最小-最大值)为9(0-38),SLICC/ACR-DI(SDI)评分的中位数(最小-最大值)为1(1-5)。组织病理学定义为1例霍奇金淋巴瘤,2例非霍奇金淋巴瘤,1例急性淋巴细胞白血病;4例胃肠道癌,1例舌鳞状细胞癌和1例肛门癌;1 例乙状结肠腺癌和 1 例胃类癌;3 例生殖器恶性肿瘤,包括 1 例外阴癌、1 例宫颈癌和 1 例外阴和宫颈癌;1 例中枢神经系统少突胶质细胞瘤;以及 1 例睾丸生殖细胞畸胎瘤。结论在一项多中心的系列研究中,狼疮随访期间的恶性肿瘤发生率估计为0.7%。在诊断出恶性肿瘤时,疾病活动度和累积损害评分的中位数都很高;考虑到成人系列中的报告,这一数字也很高。
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Childhood-onset systemic lupus erythematosus (cSLE) and malignancy: a nationwide multicentre series review.

Background: Increased malignancy frequency is well documented in adult-systemic lupus erythematosus (SLE), but with limited reports in childhood-onset SLE (cSLE) series. We explored the frequency of malignancy associated with cSLE, describing clinical and demographic characteristics, disease activity and cumulative damage, by the time of malignancy diagnosis.

Method: A retrospective case-notes review, in a nationwide cohort from 27 Pediatric Rheumatology centres, with descriptive biopsy-proven malignancy, disease activity/damage accrual, and immunosuppressive treatment were compiled in each participating centre, using a standard protocol.

Results: Of the 1757 cSLE cases in the updated cohort, 12 (0.7%) developed malignancy with median time 10 years after cSLE diagnosis. There were 91% females, median age at cSLE diagnosis 12 years, median age at malignancy diagnosis 23 years. Of all diagnosed malignancies, 11 were single-site, and a single case with concomitant multiple sites; four had haematological (0.22%) and 8 solid malignancy (0.45%). Median (min-max) SLEDAI-2 K scores were 9 (0-38), median (min-max) SLICC/ACR-DI (SDI) score were 1 (1-5) Histopathology defined 1 Hodgkin's lymphoma, 2 non-Hodgkin's lymphoma, 1 acute lymphoblastic leukaemia; 4 gastrointestinal carcinoma, 1 squamous cell carcinoma of the tongue and 1 anal carcinoma; 1 had sigmoid adenocarcinoma and 1 stomach carcinoid; 3 had genital malignancy, being 1 vulvae, 1 cervix and 1 vulvae and cervix carcinomas; 1 had central nervous system oligodendroglioma; and 1 testicle germ cell teratoma.

Conclusion: Estimated malignancy frequency of 0.7% was reported during cSLE follow up in a multicentric series. Median disease activity and cumulative damage scores, by the time of malignancy diagnoses, were high; considering that reported in adult series.

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ACS Applied Bio Materials
ACS Applied Bio Materials Chemistry-Chemistry (all)
CiteScore
9.40
自引率
2.10%
发文量
464
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