辅助生殖技术后出生儿童的朗格汉斯细胞组织细胞增生症。

IF 3.7 2区医学 Q1 OBSTETRICS & GYNECOLOGY

Reproductive biomedicine online Pub Date : 2024-07-19 DOI:10.1016/j.rbmo.2024.104379

Carrie L. Williams , Kathryn J. Bunch , Charles Stiller , Michael F.G. Murphy , Beverley J. Botting , Melanie C. Davies , Barbara Luke , Philip J. Lupo , Alastair G. Sutcliffe

{"title":"辅助生殖技术后出生儿童的朗格汉斯细胞组织细胞增生症。","authors":"Carrie L. Williams , Kathryn J. Bunch , Charles Stiller , Michael F.G. Murphy , Beverley J. Botting , Melanie C. Davies , Barbara Luke , Philip J. Lupo , Alastair G. Sutcliffe","doi":"10.1016/j.rbmo.2024.104379","DOIUrl":null,"url":null,"abstract":"<div><h3>Research question</h3><div>Are children born after assisted reproductive technology (ART) at higher risk of developing Langerhans cell histiocytosis (LCH)?</div></div><div><h3>Design</h3><div>Records of children born after ART recorded by the UK Human Fertilisation & Embryology Authority were linked to National Registry of Childhood Tumours records to determine the number of children developing LCH. Calculated person-years at risk were used in conjunction with the incidence of LCH in the general population to determine the expected number of cases if the cohort had the same incidence as the general population with similar age and sex, over the same calendar years. The standardized incidence ratio (SIR) was derived as the ratio of observed to expected cases. Exact 95% CI were calculated.</div></div><div><h3>Results</h3><div>In total, 118,155 children born after ART contributed 796,633 person-years follow-up (average follow-up 6.74 years). Eight cases of LCH were identified, compared with 3.75 cases expected (SIR 2.135, 95% CI 0.92–4.21; <em>P</em> = 0.074). Significantly more cases were associated with intracytoplasmic sperm injection (ICSI) (SIR 4.02, 95% CI 1.31–9.39) and male factor infertility (SIR 5.41, 95% CI 1.47–13.84). Most cases of LCH had single-system disease (<em>n</em> = 6).</div></div><div><h3>Conclusions</h3><div>This study found that significantly more cases of LCH were identified in children born after ICSI and in children whose parents had male factor infertility. A non-significant excess of cases in children born after ART was identified. Absolute excess risk was small. Given the rarity of LCH and the small number of cases included in this large cohort, further studies into the risk of LCH in children born after ART are indicated.</div></div>","PeriodicalId":21134,"journal":{"name":"Reproductive biomedicine online","volume":null,"pages":null},"PeriodicalIF":3.7000,"publicationDate":"2024-07-19","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"0","resultStr":"{\"title\":\"Langerhans cell histiocytosis in children born after assisted reproductive technology\",\"authors\":\"Carrie L. Williams , Kathryn J. Bunch , Charles Stiller , Michael F.G. Murphy , Beverley J. Botting , Melanie C. Davies , Barbara Luke , Philip J. Lupo , Alastair G. Sutcliffe\",\"doi\":\"10.1016/j.rbmo.2024.104379\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<div><h3>Research question</h3><div>Are children born after assisted reproductive technology (ART) at higher risk of developing Langerhans cell histiocytosis (LCH)?</div></div><div><h3>Design</h3><div>Records of children born after ART recorded by the UK Human Fertilisation & Embryology Authority were linked to National Registry of Childhood Tumours records to determine the number of children developing LCH. Calculated person-years at risk were used in conjunction with the incidence of LCH in the general population to determine the expected number of cases if the cohort had the same incidence as the general population with similar age and sex, over the same calendar years. The standardized incidence ratio (SIR) was derived as the ratio of observed to expected cases. Exact 95% CI were calculated.</div></div><div><h3>Results</h3><div>In total, 118,155 children born after ART contributed 796,633 person-years follow-up (average follow-up 6.74 years). Eight cases of LCH were identified, compared with 3.75 cases expected (SIR 2.135, 95% CI 0.92–4.21; <em>P</em> = 0.074). Significantly more cases were associated with intracytoplasmic sperm injection (ICSI) (SIR 4.02, 95% CI 1.31–9.39) and male factor infertility (SIR 5.41, 95% CI 1.47–13.84). Most cases of LCH had single-system disease (<em>n</em> = 6).</div></div><div><h3>Conclusions</h3><div>This study found that significantly more cases of LCH were identified in children born after ICSI and in children whose parents had male factor infertility. A non-significant excess of cases in children born after ART was identified. Absolute excess risk was small. Given the rarity of LCH and the small number of cases included in this large cohort, further studies into the risk of LCH in children born after ART are indicated.</div></div>\",\"PeriodicalId\":21134,\"journal\":{\"name\":\"Reproductive biomedicine online\",\"volume\":null,\"pages\":null},\"PeriodicalIF\":3.7000,\"publicationDate\":\"2024-07-19\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Reproductive biomedicine online\",\"FirstCategoryId\":\"3\",\"ListUrlMain\":\"https://www.sciencedirect.com/science/article/pii/S1472648324005686\",\"RegionNum\":2,\"RegionCategory\":\"医学\",\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"\",\"PubModel\":\"\",\"JCR\":\"Q1\",\"JCRName\":\"OBSTETRICS & GYNECOLOGY\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Reproductive biomedicine online","FirstCategoryId":"3","ListUrlMain":"https://www.sciencedirect.com/science/article/pii/S1472648324005686","RegionNum":2,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q1","JCRName":"OBSTETRICS & GYNECOLOGY","Score":null,"Total":0}

引用次数: 0

摘要

研究问题辅助生殖技术（ART）后出生的儿童患朗格汉斯细胞组织细胞增生症（LCH）的风险是否更高？设计将英国人类受精与胚胎学管理局记录的辅助生殖技术后出生的儿童记录与全国儿童肿瘤登记记录进行链接，以确定患 LCH 的儿童人数。将计算出的风险年数与普通人群的LCH发病率结合使用，以确定如果在相同的日历年中，队列的发病率与年龄和性别相似的普通人群的发病率相同，那么预计的病例数。标准化发病率（SIR）是观察病例与预期病例的比率。结果在抗逆转录病毒疗法后出生的 118,155 名儿童共接受了 796,633 人年的随访（平均随访时间为 6.74 年）。共发现 8 例 LCH，而预期为 3.75 例（SIR 2.135，95% CI 0.92-4.21；P = 0.074）。与卵胞浆内单精子注射（ICSI）（SIR 4.02，95% CI 1.31-9.39）和男性因素不育（SIR 5.41，95% CI 1.47-13.84）相关的病例明显增多。结论本研究发现，经卵胞浆内单精子显微注射（ICSI）后出生的儿童和父母患有男性因素不育症的儿童中发现的 LCH 病例明显较多。在人工授精后出生的儿童中，发现的LCH病例数明显偏高。绝对超额风险很小。鉴于LCH的罕见性和这一大型队列中包含的病例数较少，有必要进一步研究抗逆转录病毒疗法后出生的儿童患LCH的风险。

本文章由计算机程序翻译，如有差异，请以英文原文为准。

查看原文本刊更多论文

Langerhans cell histiocytosis in children born after assisted reproductive technology

Research question

Are children born after assisted reproductive technology (ART) at higher risk of developing Langerhans cell histiocytosis (LCH)?

Design

Records of children born after ART recorded by the UK Human Fertilisation & Embryology Authority were linked to National Registry of Childhood Tumours records to determine the number of children developing LCH. Calculated person-years at risk were used in conjunction with the incidence of LCH in the general population to determine the expected number of cases if the cohort had the same incidence as the general population with similar age and sex, over the same calendar years. The standardized incidence ratio (SIR) was derived as the ratio of observed to expected cases. Exact 95% CI were calculated.

Results

In total, 118,155 children born after ART contributed 796,633 person-years follow-up (average follow-up 6.74 years). Eight cases of LCH were identified, compared with 3.75 cases expected (SIR 2.135, 95% CI 0.92–4.21; P = 0.074). Significantly more cases were associated with intracytoplasmic sperm injection (ICSI) (SIR 4.02, 95% CI 1.31–9.39) and male factor infertility (SIR 5.41, 95% CI 1.47–13.84). Most cases of LCH had single-system disease (n = 6).

Conclusions

This study found that significantly more cases of LCH were identified in children born after ICSI and in children whose parents had male factor infertility. A non-significant excess of cases in children born after ART was identified. Absolute excess risk was small. Given the rarity of LCH and the small number of cases included in this large cohort, further studies into the risk of LCH in children born after ART are indicated.

求助全文

通过发布文献求助，成功后即可免费获取论文全文。去求助

来源期刊

Reproductive biomedicine online 医学-妇产科学

CiteScore

7.20

自引率

7.50%

发文量

391

审稿时长

50 days

期刊介绍： Reproductive BioMedicine Online covers the formation, growth and differentiation of the human embryo. It is intended to bring to public attention new research on biological and clinical research on human reproduction and the human embryo including relevant studies on animals. It is published by a group of scientists and clinicians working in these fields of study. Its audience comprises researchers, clinicians, practitioners, academics and patients. Context: The period of human embryonic growth covered is between the formation of the primordial germ cells in the fetus until mid-pregnancy. High quality research on lower animals is included if it helps to clarify the human situation. Studies progressing to birth and later are published if they have a direct bearing on events in the earlier stages of pregnancy.