Ratnayake Gowri M, Shekhda Kalyan Mansukhbhai, Glover Thomas, Al-Obudi Yasser, Hayes Aimee, Armonis Panagiotis, Mandair Dalvinder, Khoo Bernard, Luong TuVinh, Toumpanakis Christos, Grossman Ashley, Caplin Martyn
Neuroendocrine Tumour Unit, ENETS Centre of Excellence, Royal Free Hospital, London, UK.
Department of Endocrinology and Diabetes, University College London Hospitals, London, UK.
J Neuroendocrinol. 2025 Jan;37(1):e13465. doi: 10.1111/jne.13465. Epub 2024 Nov 6.
Neuroendocrine neoplasms (NENs) arise from the diffuse endocrine system and have been considered to be rare. However, the incidence and prevalence of these tumours have increased in recent years, and they are being seen in younger patients including women in the reproductive age group. Due to the paucity of data, diagnostic and therapeutic strategies in managing such tumours during pregnancy can be challenging to both treating physicians and patients. This article describes the experience and outcomes of managing pregnant women with NEN at a European Neuroendocrine Tumour Society (ENETS) Centre of Excellence. In this retrospective analysis, we evaluated a total of 22 pregnancies in 18 pregnant women with concurrent diagnoses of NENs who were managed at Royal Free Hospital ENETS Centre of Excellence throughout their pregnancy. These were identified from our tumour registry of 3500 NEN patients between 2015 and 2023. Cross-sectional imaging (computed tomography (CT)/magnetic resonance imaging (MRI)), pre- and post-pregnancy, for each patient was reviewed by an experienced radiologist. Tumour growth rate (TGR) was calculated using the formula: TGR = 100 × [exp (TG) - 1]; TG. [3 × log (D2/D1)]/time (months), where D1 is the tumour size at date 1; D2 is the tumour size at date 2; and time (months) = (Date 2 - Date 1 + 1)/30.44. Tumour growth rate pre-conception (TGRpc) and tumour growth rate post-partum (TGRpp) were calculated for each patient. In a sub-group of patients, positivity for oestrogen and progesterone receptors were analysed on the tumour tissue to evaluate whether the presence of these receptors affected tumour progression during the pregnancy. We also reviewed the pregnancy outcome in patients treated with somatostatin analogues during pregnancy. We analysed the data of a total 22 pregnancy encounters in 18 women: 15 pregnancies (68%) preceded the diagnosis of the NEN, whereas the diagnosis of NEN was made during pregnancy or in the post-partum period in 5 (23%) and 2 (9%) pregnancies respectively. Eight patients (44%) had a diagnosis of a pancreatic NEN, whereas 5 (28%) were diagnosed with mid-gut NENs, and a further 5 at other sites. The majority of the patients (n = 12, 67%) had evidence of metastatic disease at the time of diagnosis. Most pregnancies had a successful outcome (n = 19, 86%), whereas 3 patients (14%) had miscarriages in the 1st trimester. Five patients in total of 6 pregnancies were treated with somatostatin analogues as monotherapy during the pregnancy, and all of them had stable disease after pregnancy. All of them delivered healthy babies without any side effects or complications due to therapy. The average TGRpc was -0.8% (n = 5) and the average TGRpp was +0.96% (n = 6); 2 patients who did not have suitable targets for calculation of TGRpc developed new lesions suggesting disease progression. Moreover, 2 of the 4 patients who have had both pre-conception and post-pregnancy scans showed an increase in TGRpp compared to TGRpc. The management of NENs during pregnancy should be multidisciplinary with an individualised approach to each patient. Somatostatin analogues appear to be safe during pregnancy, though further robust studies are needed. Pregnancy per se may accelerate tumour progression, and patients should be counselled regarding this possibility.
神经内分泌肿瘤(NENs)起源于弥散内分泌系统,过去一直被认为较为罕见。然而,近年来这些肿瘤的发病率和患病率有所上升,在包括育龄期女性在内的年轻患者中也有发现。由于数据有限,在孕期管理此类肿瘤的诊断和治疗策略对治疗医生和患者来说都具有挑战性。本文介绍了在欧洲神经内分泌肿瘤学会(ENETS)卓越中心管理患有NEN的孕妇的经验和结果。在这项回顾性分析中,我们评估了18名同时诊断为NEN的孕妇的22次妊娠情况,这些孕妇在皇家自由医院ENETS卓越中心整个孕期都接受了管理。这些病例是从我们2015年至2023年的3500例NEN患者肿瘤登记中识别出来的。由一位经验丰富的放射科医生对每位患者孕前和产后的横断面成像(计算机断层扫描(CT)/磁共振成像(MRI))进行了复查。肿瘤生长率(TGR)使用以下公式计算:TGR = 100 × [exp(TG) - 1];TG = [3 × log(D2/D1)]/时间(月),其中D1是第1次检查时的肿瘤大小;D2是第2次检查时的肿瘤大小;时间(月)=(日期2 - 日期1 + 1)/30.44。计算了每位患者的孕前肿瘤生长率(TGRpc)和产后肿瘤生长率(TGRpp)。在一组患者中,对肿瘤组织进行了雌激素和孕激素受体阳性分析,以评估这些受体的存在是否会影响孕期肿瘤进展。我们还回顾了孕期接受生长抑素类似物治疗的患者的妊娠结局。我们分析了18名女性共22次妊娠的数据:15次妊娠(68%)在NEN诊断之前,而分别有5次(23%)和2次(9%)妊娠在孕期或产后诊断出NEN。8名患者(44%)诊断为胰腺NEN,5名(28%)诊断为中肠NEN,另有5名在其他部位。大多数患者(n = 12,67%)在诊断时已有转移性疾病证据。大多数妊娠结局成功(n = 19,86%),而3名患者(14%)在孕早期流产。6次妊娠中的5名患者在孕期接受了生长抑素类似物单药治疗,所有患者产后疾病稳定。她们均分娩出健康婴儿,且未出现因治疗导致的任何副作用或并发症。平均TGRpc为 -0.8%(n = 5),平均TGRpp为 +0.96%(n = 6);2名没有适合计算TGRpc的靶点的患者出现了新病灶,提示疾病进展。此外,4名进行了孕前和产后扫描的患者中有2名显示TGRpp较TGRpc有所增加。孕期NEN的管理应采取多学科、针对每位患者的个体化方法。生长抑素类似物在孕期似乎是安全的,不过还需要进一步的有力研究。妊娠本身可能会加速肿瘤进展,应就此可能性向患者提供咨询。