Williams Carrie L, Bunch Kathryn J, Stiller Charles, Murphy Michael F G, Botting Beverley J, Davies Melanie C, Luke Barbara, Lupo Philip J, Sutcliffe Alastair G
UCL Great Ormond Street Institute of Child Health, London, UK.
Childhood Cancer Research Group, Department of Paediatrics, University of Oxford, Oxford, UK.
Reprod Biomed Online. 2024 Dec;49(6):104379. doi: 10.1016/j.rbmo.2024.104379. Epub 2024 Jul 19.
Are children born after assisted reproductive technology (ART) at higher risk of developing Langerhans cell histiocytosis (LCH)?
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.
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).
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.
通过辅助生殖技术（ART）出生的儿童患朗格汉斯细胞组织细胞增多症（LCH）的风险是否更高？
英国人类受精与胚胎管理局记录的ART出生儿童记录与国家儿童肿瘤登记处记录相链接，以确定患LCH的儿童数量。计算出的风险人年数与一般人群中LCH的发病率相结合，以确定如果该队列在相同日历年份中具有与年龄和性别相似的一般人群相同发病率时的预期病例数。标准化发病率比（SIR）作为观察到的病例数与预期病例数的比值得出。计算出确切的95%置信区间。
共有118,155名通过ART出生的儿童提供了796,633人年的随访（平均随访6.74年）。确定了8例LCH病例，而预期为3.75例（SIR 2.135，95%置信区间0.92 - 4.21；P = 0.074）。显著更多的病例与胞浆内单精子注射（ICSI）（SIR 4.02，95%置信区间1.31 - 9.39）和男性因素不育（SIR 5.41，95%置信区间1.47 - 13.84）相关。大多数LCH病例为单系统疾病（n = 6）。
本研究发现，ICSI出生的儿童以及父母有男性因素不育的儿童中，LCH病例显著更多。在ART出生的儿童中发现病例有非显著的超额情况。绝对超额风险较小。鉴于LCH的罕见性以及该大型队列中纳入的病例数量较少，表明需要进一步研究ART出生儿童患LCH的风险。
