Kruseová Jarmila, Gottfriedová Barbora, Zichová Andrea, Švojgr Karel, Hošek Petr, Lukš Aleš, Kynčl Martin, Eckschlager Tomáš
Department of Paediatric Haematology and Oncology, 2nd Faculty of Medicine, Charles University and Motol University Hospital, Prague, Czech Republic.
Department of Radiology, 2nd Faculty of Medicine, Charles University and Motol University Hospital, Prague, Czech Republic.
Clin Epidemiol. 2021 Aug 11;13:707-716. doi: 10.2147/CLEP.S317903. eCollection 2021.
Cancer treatment can cause various long-term side effects, including those that impact ultrasound findings. During follow-up of childhood cancer survivors (CCSs), we often detected sporadic renal angiomyolipomas without histological confirmation (SAMLs), which is why we initiated this study. We compared the occurrence of SAML in CCSs to the previously reported data from a non-cancer population and correlated SAML with cancer treatment-related factors.
The cohort included 1098 CCSs (median age at cancer diagnosis (dg) 4.3 years) who had ultrasound follow-up (2014-2019). Of the CCSs, 525 (48%) were female, 132 (12%) had subsequent neoplasms (SNs), and 110 (10%) had genetic syndromes. CCSs were treated for lymphomas 269 (24%) and solid tumors 829 (76%). None of the CCSs had tuberous sclerosis complex (TSC).
SAML developed in 48 (4.4%) CCSs; of these, 20 (42%) had SNs. The coincidence of SAMLs and SNs was found in CCSs with a follow-up period exceeding 20 years. The median age at SAML dg was 27.9 years (interquartile range (IQR) 22.3-34.1), and the median time to SAML dg was 22.6 years (IQR 17.4-27.6). Twenty-one (44%) CCSs developed multiple or bilateral SAMLs lesions; of these, six (12%) were in the radiotherapy field. SAML occurrence correlated with radiotherapy of the retroperitoneum (1.65-fold higher with 95% CI 0.90-3.02). The correlations with other cancer treatment factors and with female sex were less clear.
This study revealed the occurrence of SAMLs in CCSs to be 10 times higher than that in non-cancer studies. The current characteristics of CCSs with SAMLs: younger age, and more bilateral or multiple lesions are more similar to TSC associated angiomyolipoma. Moreover, we observed a coincidence of SAMLs with SNs. Our results support the hypothesis that SAML development in CCSs is not simply a late effect of therapy, and indicates other factors are involved in SAML development.
癌症治疗可导致各种长期副作用,包括影响超声检查结果的副作用。在儿童癌症幸存者(CCSs)的随访过程中,我们经常检测到未经组织学证实的散发性肾血管平滑肌脂肪瘤(SAMLs),这就是我们开展这项研究的原因。我们将CCSs中SAML的发生率与先前报道的非癌症人群的数据进行了比较,并将SAML与癌症治疗相关因素进行了关联分析。
该队列包括1098名接受超声随访(2014 - 2019年)的CCSs(癌症诊断时的中位年龄为4.3岁)。在这些CCSs中,525名(48%)为女性,132名(12%)有后续肿瘤(SNs),110名(10%)有遗传综合征。CCSs中269名(24%)接受淋巴瘤治疗,829名(76%)接受实体瘤治疗。所有CCSs均无结节性硬化症(TSC)。
48名(4.4%)CCSs发生了SAML;其中20名(42%)有SNs。在随访期超过20年的CCSs中发现了SAMLs与SNs的巧合情况。SAML诊断时的中位年龄为27.9岁(四分位间距(IQR)为22.3 - 34.1),SAML诊断的中位时间为22.6年(IQR为17.4 - 27.6)。21名(44%)CCSs出现了多发或双侧SAMLs病变;其中6名(12%)位于放疗区域。SAML的发生与腹膜后放疗相关(高1.65倍,95%置信区间为0.90 - 3.02)。与其他癌症治疗因素以及与女性性别的相关性不太明确。
本研究显示CCSs中SAML的发生率比非癌症研究中的高出10倍。具有SAMLs的CCSs的当前特征:年龄较小,且更多双侧或多发病变,更类似于TSC相关的血管平滑肌脂肪瘤。此外,我们观察到SAMLs与SNs的巧合情况。我们的结果支持这样一种假设:CCSs中SAML的发生不仅仅是治疗的晚期效应,并且表明其他因素参与了SAML的发生。