The systematic research was conducted from January 2000 to December 2021 through MEDLINE via PubMed, Cochrane Library, and Embase databases. Two separate meta-analyses were performed for PTC and FTC.
After the selection phase, a total of 15 studies (3,332 screened) met the inclusion criteria and are reported in the present systematic review. Five studies were conducted in Europe, five in North America, two in South America, one in Asia, one reported data for 49 countries and territories across the five continents, and one from both the USA and Africa. Most of the studies (n = 14) reported data obtained from national registries, and only one provided information collected from hospital medical records. Beyond the actual trend over time, our study reported a pooled global incidence rate (IR) of PTC and FTC in the pediatric age of 0.46 (95% CI: 0.33-0.59) and 0.07 (95% CI: 0.02-0.12) per 100,000 person-years, respectively. The highest IRs were recorded among Caucasian girls, and the lowest in black or other races/ethnicities.
Our data confirm that DTC in the pediatric population is a rare condition. The pooled IRs of the studies included in this meta-analysis are ~0.5 for PTC, which is the most common histological type when both genders and all age groups are considered. The implementation of a prospective international registry on pediatric DTC, as part of the wider European Registries for Rare Endocrine Conditions, has been recently proposed. In addition to providing relevant information on the clinical behavior of this rare disease, standardization of data collection will be pivotal to fill current gaps and allow an accurate estimation of the real incidence and risk factors of DTC.
■系统研究于2000年1月至2021年12月通过MEDLINE通过PubMed进行,科克伦图书馆,和Embase数据库。对PTC和FTC进行了两个单独的荟萃分析。
■在选择阶段之后,共有15项研究(3,332项筛选)符合纳入标准,并在本系统综述中报告.在欧洲进行了五项研究,五个在北美,两个在南美洲,一个在亚洲,一个报告了五大洲49个国家和地区的数据,一个来自美国和非洲。大多数研究(n=14)报告了从国家注册管理机构获得的数据,只有一个提供了从医院医疗记录中收集的信息。除了随着时间的推移的实际趋势,我们的研究报告了儿童年龄的PTC和FTC的合并全球发病率(IR)为每100,000人年0.46(95%CI:0.33-0.59)和0.07(95%CI:0.02-0.12),分别。最高的IRs记录在高加索女孩中,在黑人或其他种族/民族中最低。
■我们的数据证实,儿科人群中的DTC是一种罕见的疾病。本荟萃分析中包含的研究的合并IR为PTC的约0.5,当考虑到性别和所有年龄组时,这是最常见的组织学类型。关于儿科DTC的前瞻性国际注册的实施,作为更广泛的欧洲罕见内分泌疾病登记处的一部分,最近有人提出。除了提供有关这种罕见疾病的临床行为的相关信息外,数据收集的标准化对于填补当前空白并准确估计DTC的实际发生率和风险因素至关重要。