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Invasive Acantholytic Extramammary Paget's Disease of the Vulva With Mammary Carcinoma-Like Differentiation and MMR Loss: Report of an Unusual Case and Its Clinical Mimics 外阴浸润性棘溶解性乳腺外佩吉特病伴乳腺癌样分化和MMR丢失:1例罕见病例及其临床模拟报告
IF 1.3 Q3 OBSTETRICS & GYNECOLOGY Pub Date : 2025-12-15 DOI: 10.1016/j.gore.2025.102007
Jessica Claus , Janina Pearce , Areta Bojko , Padmini Manrai , M.Ruhul Quddus , Shivali Marketkar

Background

Paget’s disease (EMPD) of the vulva primarily affects postmenopausal women and can present significant challenges in early clinical detection.
Case Presentation: We report an exceedingly rare case of invasive acantholytic extramammary Paget’s disease in a 78-year-old female patient. After biopsy-proven carcinoma, a radical hemivulvectomy of the left vulva was performed, which revealed an invasive adenocarcinoma, most consistent with primary extramammary Paget’s carcinoma. Histologic examination demonstrated a unique morphology, featuring an in-situ component with suprabasal intraepidermal acantholysis and an invasive component with various morphologies, including basaloid, skin adnexal-like, focal squamoid, and mammary-like differentiation, resembling ductal carcinoma in situ (DCIS) with comedonecrosis and invasive ductal carcinoma. This varied morphology has not been described before. Interestingly, the tumor shows loss of MMR protein (MSH2, MSH6).

Conclusion

In the literature, acantholytic Paget’s disease and acantholytic anaplastic Paget’s disease have been used interchangeably. While anaplastic Paget’s disease has been reported in the mammary and extramammary sites (scrotum, esophagus), involvement of the vulva has been reported only once in the literature. However, the current case is the first documented loss of MMR. Both cases presented with invasive disease ranging in depth from 3-5 mm, and positive lymph nodes emphasizing on the urgent need to further explore prognostic factors of this rare entity and to differentiate it from other acantholytic lesions, such as squamous cell carcinoma in situ and other acantholytic dermatoses.
外阴佩吉特病(EMPD)主要影响绝经后妇女,在早期临床检测中存在重大挑战。病例介绍:我们报告一位78岁的女性患者,患极罕见的侵袭性乳腺外棘突溶解性佩吉特病。经活检证实为癌后,行左外阴根治性半外阴切除术,发现为浸润性腺癌,与原发性乳腺外佩吉特癌最一致。组织学检查显示其独特的形态,具有基底上表皮内棘层溶解的原位成分和具有多种形态的浸润性成分,包括基底样、皮肤附件样、局灶鳞状和乳腺样分化,类似于导管原位癌(DCIS)伴粉刺坏死和浸润性导管癌。这种不同的形态以前没有被描述过。有趣的是,肿瘤显示MMR蛋白(MSH2, MSH6)的缺失。结论在文献中,棘溶性Paget病和棘溶性间变性Paget病被交替使用。虽然间变性Paget病在乳腺和乳腺外部位(阴囊、食道)有报道,但在文献中仅报道过一次外阴受累。然而,目前的病例是首次记录的MMR损失。两例均表现为浸润性疾病,深度为3-5 mm,淋巴结阳性,强调迫切需要进一步探讨这种罕见实体的预后因素,并将其与其他棘囊性病变(如原位鳞状细胞癌和其他棘囊性皮肤病)区分开来。
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引用次数: 0
Treatment delay and prolongation in locally advanced cervical cancer 局部晚期子宫颈癌的治疗延迟和延长
IF 1.3 Q3 OBSTETRICS & GYNECOLOGY Pub Date : 2025-12-13 DOI: 10.1016/j.gore.2025.102004
Maya Gross , Joyce Y. Wang , Barbara A. Goff , Ting Martin Ma , Kylie H. Kang , Kemi M. Doll , Soledad Jorge

Objectives

Prolonged chemoradiation (CRT) duration predicts inferior survival in locally advanced cervical cancer (LACC). Pre-treatment delays and causes of prolonged treatment duration are less characterized. We aimed to quantify delays across the care continuum and identify predictors of delayed CRT initiation and treatment prolongation.

Methods

In this retrospective cohort study, we evaluated LACC patients receiving definitive CRT from 2013 to 2024 at an NCI-designated Cancer Center. We assessed care intervals and sociodemographic/clinical variables. Primary outcome was total patient delay (diagnosis to CRT completion), subdivided into delayed treatment initiation (>75th percentile from diagnosis to CRT) and prolonged CRT duration (>56 days). Secondary outcomes included predictors of delay / prolongation (x2 tests, logistic regression), progression free survival (PFS), and overall survival (OS).

Results

Among 92 patients, median time to CRT initiation was 49 days (IQR 40–62) and median treatment duration was 57 days (IQR 52–60). Treatment delay or prolongation was associated with insurance type, external beam radiation therapy (EBRT) at an outside facility, and adenocarcinoma histology (p < 0.05). Time from diagnosis to completion of LACC staging was the interval most predictive of treatment delay (OR 3.7, CI 1.8–7.7). Treatment prolongation was associated with longer intervals between EBRT and brachytherapy (BT) (OR 2.6,CI 1.6–4.2) but not with time to initiate EBRT or EBRT duration. PFS and OS were not associated with primary outcomes.

Conclusions

A quarter of patients waited over 2 months to initiate CRT, and half experienced prolonged treatment duration. Delays in staging and transitions in care, especially between institutions, contributed significantly to total patient delay.
目的探讨局部晚期宫颈癌(LACC)放化疗时间延长的预后。治疗前的延迟和治疗持续时间延长的原因较少被描述。我们的目的是量化整个护理连续体的延迟,并确定延迟CRT开始和治疗延长的预测因素。方法在这项回顾性队列研究中,我们评估了2013年至2024年在nci指定的癌症中心接受最终CRT的LACC患者。我们评估了护理间隔和社会人口统计学/临床变量。主要终点是患者总延迟(诊断到CRT完成),再细分为延迟治疗开始(从诊断到CRT的第75个百分位数)和延长CRT持续时间(56天)。次要结局包括延迟/延长(x2检验,逻辑回归)、无进展生存期(PFS)和总生存期(OS)的预测因子。结果92例患者中位开始CRT的时间为49天(IQR 40-62),中位治疗时间为57天(IQR 52-60)。治疗延迟或延长与保险类型、外部设施外束放射治疗(EBRT)和腺癌组织学相关(p < 0.05)。从诊断到完成LACC分期的时间是最能预测治疗延迟的时间间隔(OR 3.7, CI 1.8-7.7)。治疗延长与EBRT和近距离治疗(BT)之间的间隔时间延长相关(OR 2.6,CI 1.6-4.2),但与开始EBRT的时间或EBRT持续时间无关。PFS和OS与主要结局无关。结论四分之一的患者开始CRT的时间超过2个月,一半的患者治疗时间延长。延迟的分期和过渡的护理,特别是机构之间,贡献了显著的总患者延误。
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引用次数: 0
Prognostic and therapeutic implications of clinical-radiologic discrepancy in parametrial invasion prior to primary radical hysterectomy in cervical cancer 宫颈癌原发性根治性子宫切除术前参数浸润的临床-放射差异对预后和治疗的意义
IF 1.3 Q3 OBSTETRICS & GYNECOLOGY Pub Date : 2025-12-09 DOI: 10.1016/j.gore.2025.102002
Ester P. Olthof , Nicholai A. Oostveen , Maaike A. van der Aa , Ruud L.M. Bekkers , Constantijne H. Mom , Jacobus van der Velden , Joost Nederend , Edith M.G. van Esch

Aim

This study evaluates the prognostic and therapeutic implications of clinical-radiologic discrepancy in parametrial invasion of cervical cancer prior to radical hysterectomy. We compared patients with radiological presence but clinical absence of parametrial invasion (discrepancy group) to those without radiologic and clinical suspicion of parametrial invasion (consensus group).

Methods

Women with International Federation of Gynaecology and Obstetrics (2009) stage IA-IIA cervical cancer, diagnosed between 2009 and 2017, who underwent magnetic resonance imaging prior to radical hysterectomy were retrospectively selected from the Netherlands Cancer Registry. Kaplan-Meier estimates and Cox proportional hazards were used for survival and logistic regression for risk of adjuvant therapy and toxicity.

Results

Of 886 patients included, 87 (10%) had clinical-radiologic parametrial invasion discrepancy. Patients with discrepancy were more likely to have poor prognostic factors (i.e., a larger tumor, increased depth of invasion, lymphovascular space invasion, nodal metastases and positive resection margins) than those without. The 5-year disease-free and overall survival rates were lower in the discrepancy (74% and 82%) than in the consensus group (86% and 92%). However, after confounder adjustments, disease-free and overall survival were not affected by clinical-radiologic discrepancy. Patients with discrepancy in parametrial invasion were more likely to receive adjuvant therapy (54% vs 23%) and experience therapy-related toxicity (44% vs 29%).

Conclusion

Clinical-radiologic discrepancy of parametrial invasion occurs in approximately 10% of patients and is associated with poor prognostic factors and increased likelihood of adjuvant therapy and toxicity. This highlights the importance of addressing these factors in treatment counselling for either primary chemoradiotherapy or surgery.
目的探讨宫颈癌根治性子宫切除术前参数浸润的临床-放射学差异对预后和治疗的意义。我们比较了影像学上存在但临床没有参数性侵犯的患者(差异组)和没有影像学和临床怀疑参数性侵犯的患者(共识组)。方法回顾性选择2009年至2017年期间诊断为国际妇产科联合会(2009年)IA-IIA期宫颈癌的妇女,并在根治性子宫切除术前接受磁共振成像。Kaplan-Meier估计和Cox比例风险用于生存和辅助治疗风险和毒性的逻辑回归。结果886例患者中,87例(10%)存在临床-放射参数侵袭差异。与无差异的患者相比,有差异的患者更有可能存在不良预后因素(即肿瘤较大、浸润深度增加、淋巴血管间隙浸润、淋巴结转移和切除边缘阳性)。差异组的5年无病生存率和总生存率(74%和82%)低于共识组(86%和92%)。然而,在混杂因素调整后,无病生存和总生存不受临床-放射差异的影响。参数浸润差异的患者更有可能接受辅助治疗(54%对23%),并经历治疗相关的毒性(44%对29%)。结论约10%的患者出现参数性侵袭的临床-影像学差异,这与预后不良因素、辅助治疗的可能性和毒性增加有关。这突出了在初级放化疗或手术治疗咨询中解决这些因素的重要性。
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引用次数: 0
Abdominal wall blocks in robotic hysterectomy for endometrial cancer are associated with a modest reduction in the frequency of patients receiving post-operative intravenous opioids 子宫内膜癌机器人子宫切除术中腹壁阻滞与术后静脉注射阿片类药物的患者频率适度降低相关
IF 1.3 Q3 OBSTETRICS & GYNECOLOGY Pub Date : 2025-12-06 DOI: 10.1016/j.gore.2025.101995
Ioana Bondre , H. Meryem Soylu , Francesca Corbetta , Breana Hill , Elise Wilson , Rodney Gabriel , Ramez Eskander , Michael McHale , Cheryl Saenz , Pratibha Binder , Steven Plaxe

Objective

To evaluate the association of abdominal wall blocks at the time of robotic hysterectomy performed for endometrial cancer (EC) with in-hospital post-operative opioids.

Methods

We performed a retrospective cohort study of patients in the Vizient database who had robotic hysterectomy for EC between January 2019 and December 2022. The exposure was any block coded separate from the surgery, and the primary outcomes were any IV/oral opioid. Fentanyl was excluded due to frequent intra-op use. Length of stay (LOS) and direct cost were compared. The association between block and opioid use was assessed via risk ratio. The quantity of each opioid was expressed in standard resource units (SRU) – a metric used to standardize the consumption of medications across facilities.

Results

Three hundred and twenty of the 9062 patients (3.5 %) had a block and 8,742 (96.5 %) did not. Blocks were performed at 66 (20.4 %) hospitals. 184 patients (57.5 %) in the block group received an IV opioid vs 5,890 (67.4 %) of the patients without a block (RR 0.85, 95 % CI 0.77–0.94, p < 0.001). Patients who received blocks were less likely to receive IV (52.5 % vs 64.8 %, RR 0.81, 95 % CI 0.73–0.89, p < 0.001) and oral hydromorphone (6.9 % vs 3.9 %, RR 2.53, 95 % CI 1.68–3.81, p < 0.001) and more likely to receive oral hydrocodone/acetaminophen (11.3 % vs 0.88 %, RR 12.77, 95 % CI 8.73–18.67, p < 0.001).

Conclusion

Blocks are infrequently performed and are associated with a modest reduction in the number of patients receiving post-op IV opioids. We found no difference in LOS or cost. Limitations include lack of detail regarding type of block, anesthetic, and phase of care.
目的探讨子宫内膜癌(EC)机器人子宫切除术时腹壁阻滞与院内阿片类药物的关系。方法:我们对Vizient数据库中2019年1月至2022年12月期间因EC接受机器人子宫切除术的患者进行了回顾性队列研究。暴露是与手术分开的,主要结果是静脉/口服阿片类药物。芬太尼因术中频繁使用被排除在外。比较住院时间(LOS)和直接费用。通过风险比评估阻滞和阿片类药物使用之间的关系。每种阿片类药物的数量以标准资源单位(SRU)表示,这是一种用于标准化各设施药物消耗的度量。结果9062例患者中有320例(3.5%)发生阻滞,8742例(96.5%)未发生阻滞。66家(20.4%)医院实施阻滞。阻断组184例患者(57.5%)接受静脉注射阿片类药物,未阻断组5890例患者(67.4%)(RR 0.85, 95% CI 0.77-0.94, p < 0.001)。接受阻断治疗的患者较少接受静脉注射(52.5% vs 64.8%, RR 0.81, 95% CI 0.73-0.89, p < 0.001)和口服氢吗啡酮(6.9% vs 3.9%, RR 2.53, 95% CI 1.68-3.81, p < 0.001),更可能接受口服氢可酮/对乙酰氨基酚(11.3% vs 0.88%, RR 12.77, 95% CI 8.73-18.67, p < 0.001)。结论阻滞很少发生,并且与术后接受静脉注射阿片类药物的患者数量适度减少有关。我们发现LOS和成本没有差异。局限性包括缺乏关于阻滞类型、麻醉和护理阶段的细节。
{"title":"Abdominal wall blocks in robotic hysterectomy for endometrial cancer are associated with a modest reduction in the frequency of patients receiving post-operative intravenous opioids","authors":"Ioana Bondre ,&nbsp;H. Meryem Soylu ,&nbsp;Francesca Corbetta ,&nbsp;Breana Hill ,&nbsp;Elise Wilson ,&nbsp;Rodney Gabriel ,&nbsp;Ramez Eskander ,&nbsp;Michael McHale ,&nbsp;Cheryl Saenz ,&nbsp;Pratibha Binder ,&nbsp;Steven Plaxe","doi":"10.1016/j.gore.2025.101995","DOIUrl":"10.1016/j.gore.2025.101995","url":null,"abstract":"<div><h3>Objective</h3><div>To evaluate the association of abdominal wall blocks at the time of robotic hysterectomy performed for endometrial cancer (EC) with in-hospital post-operative opioids.</div></div><div><h3>Methods</h3><div>We performed a retrospective cohort study of patients in the Vizient database<!--> <!-->who had robotic hysterectomy for EC between January 2019 and December 2022. The exposure was any<!--> <!-->block coded separate from the surgery, and the primary outcomes were any IV/oral opioid. Fentanyl was excluded due to frequent intra-op<!--> <!-->use. Length of stay (LOS) and<!--> <!-->direct cost<!--> <!-->were compared. The association between block and opioid use<!--> <!-->was assessed via risk ratio. The quantity of each opioid was expressed in standard resource units (SRU) – a metric<!--> <!-->used to standardize the consumption of medications across facilities.</div></div><div><h3>Results</h3><div>Three hundred and twenty of the 9062 patients (3.5 %) had a block and 8,742 (96.5 %) did not. Blocks were performed at 66 (20.4 %) hospitals.<!--> <!-->184 patients (57.5 %) in the block group received an IV opioid vs 5,890 (67.4 %) of the<!--> <!-->patients without a block (RR 0.85, 95 % CI 0.77–0.94, p &lt; 0.001). Patients who received blocks were less likely to receive IV (52.5 % vs 64.8 %, RR 0.81, 95 % CI 0.73–0.89, p &lt; 0.001)<!--> <!-->and oral hydromorphone (6.9 % vs 3.9 %, RR 2.53, 95 % CI 1.68–3.81, p &lt; 0.001)<!--> <!-->and more likely to receive oral hydrocodone/acetaminophen (11.3 % vs 0.88 %, RR 12.77, 95 % CI 8.73–18.67, p &lt; 0.001).</div></div><div><h3>Conclusion</h3><div>Blocks are infrequently performed and<!--> <!-->are associated with a modest reduction in the number of patients receiving post-op IV opioids.<!--> <!-->We found no difference<!--> <!-->in<!--> <!-->LOS or cost. Limitations include lack of detail regarding type of block, anesthetic, and phase of care.</div></div>","PeriodicalId":12873,"journal":{"name":"Gynecologic Oncology Reports","volume":"63 ","pages":"Article 101995"},"PeriodicalIF":1.3,"publicationDate":"2025-12-06","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145697958","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Mucinous ovarian neoplasm – outcomes of a rare tumor 卵巢黏液性肿瘤——一种罕见肿瘤的预后
IF 1.3 Q3 OBSTETRICS & GYNECOLOGY Pub Date : 2025-12-01 DOI: 10.1016/j.gore.2025.101996
Bahareh Hamedi , Shilpa Mokshagundam , Antonio Lembo , S John Weroha , Michaela E. McGree , Amanda L. Tapia , Carrie L. Langstraat

Background

Mucinous epithelial ovarian (mEO) tumors are rare entities. In this study, we describe the management and outcomes of patients treated for mucinous borderline tumor and mucinous adenocarcinoma of the ovary.

Methods

This was a retrospective review of patients with mEO tumors from 1988 − 2021 from a single institution database. Primary outcomes were disease recurrence and death within five years post-surgery, evaluated using Cox proportional hazards and Kaplan-Meier survival analysis.

Results

We identified 262 patients with mEO tumors with a mean age of 54.9 years. One-hundred and forty-three patients (55 %) had borderline tumors and 119 (45 %) adenocarcinoma. Most patients underwent complete surgical resection (95 %).
Those with mucinous borderline tumors had favorable prognosis, with 5-year progression-free survival (PFS) and overall survival (OS) of 97%. Among patients with mucinous adenocarcinoma, 5-year PFS was 78% and 5-year OS was 80%. Fifty-seven patients with adenocarcinoma (48%) received adjuvant chemotherapy.
In univariate analysis, adenocarcinoma histology, residual disease, advanced FIGO stage (II-IV), and receipt of adjuvant chemotherapy were associated with recurrence and death within five years of surgery. While a considerable proportion of patients in both cohorts underwent gastrointestinal (GI) endoscopy, all findings were negative/benign.

Conclusions

Mucinous tumors of the ovary have overall favorable survival outcomes. In this cohort, residual disease, histopathology, and receipt of adjuvant chemotherapy were associated with disease recurrence. In addition, GI workup was of limited utility. Further study is needed to clarify ideal adjuvant treatment and GI workup.
背景:卵巢粘液上皮(mEO)肿瘤是一种罕见的肿瘤。在这项研究中,我们描述了治疗卵巢粘液交界性肿瘤和粘液腺癌的患者的管理和结果。方法回顾性分析1988 - 2021年单一机构数据库中mEO肿瘤患者。主要结局是术后5年内疾病复发和死亡,采用Cox比例风险和Kaplan-Meier生存分析进行评估。结果262例mEO肿瘤患者,平均年龄54.9岁。143例(55%)为交界性肿瘤,119例(45%)为腺癌。大多数患者接受了完全手术切除(95%)。黏液交界性肿瘤患者预后良好,5年无进展生存期(PFS)和总生存期(OS)为97%。粘液腺癌患者的5年PFS为78%,5年OS为80%。57例腺癌患者(48%)接受了辅助化疗。在单因素分析中,腺癌组织学、残留病变、晚期FIGO分期(II-IV)和接受辅助化疗与手术后5年内的复发和死亡相关。虽然两个队列中相当比例的患者接受了胃肠道(GI)内窥镜检查,但所有结果均为阴性/良性。结论卵巢黏液性肿瘤总体生存率较好。在这个队列中,残留疾病、组织病理学和接受辅助化疗与疾病复发有关。此外,GI检查的作用有限。理想的辅助治疗和GI检查需要进一步的研究。
{"title":"Mucinous ovarian neoplasm – outcomes of a rare tumor","authors":"Bahareh Hamedi ,&nbsp;Shilpa Mokshagundam ,&nbsp;Antonio Lembo ,&nbsp;S John Weroha ,&nbsp;Michaela E. McGree ,&nbsp;Amanda L. Tapia ,&nbsp;Carrie L. Langstraat","doi":"10.1016/j.gore.2025.101996","DOIUrl":"10.1016/j.gore.2025.101996","url":null,"abstract":"<div><h3>Background</h3><div>Mucinous epithelial ovarian (mEO) tumors are rare entities. In this study, we describe the management and outcomes of patients treated for mucinous borderline tumor and mucinous adenocarcinoma of the ovary.</div></div><div><h3>Methods</h3><div>This was a retrospective review of patients with mEO tumors from 1988 − 2021 from a single institution database. Primary outcomes were disease recurrence and death within five years post-surgery, evaluated using Cox proportional hazards and Kaplan-Meier survival analysis.</div></div><div><h3>Results</h3><div>We identified 262 patients with mEO tumors with a mean age of 54.9 years. One-hundred and forty-three patients (55 %) had borderline tumors and 119 (45 %) adenocarcinoma. Most patients underwent complete surgical resection (95 %).</div><div>Those with mucinous borderline tumors had favorable prognosis, with 5-year progression-free survival (PFS) and overall survival (OS) of 97%. Among patients with mucinous adenocarcinoma, 5-year PFS was 78% and 5-year OS was 80%. Fifty-seven patients with adenocarcinoma (48%) received adjuvant chemotherapy.</div><div>In univariate analysis, adenocarcinoma histology, residual disease, advanced FIGO stage (II-IV), and receipt of adjuvant chemotherapy were associated with recurrence and death within five years of surgery. While a considerable proportion of patients in both cohorts underwent gastrointestinal (GI) endoscopy, all findings were negative/benign.</div></div><div><h3>Conclusions</h3><div>Mucinous tumors of the ovary have overall favorable survival outcomes. In this cohort, residual disease, histopathology, and receipt of adjuvant chemotherapy were associated with disease recurrence. In addition, GI workup was of limited utility. Further study is needed to clarify ideal adjuvant treatment and GI workup.</div></div>","PeriodicalId":12873,"journal":{"name":"Gynecologic Oncology Reports","volume":"62 ","pages":"Article 101996"},"PeriodicalIF":1.3,"publicationDate":"2025-12-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145620312","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Ovarian carcinoid tumor with carcinoid heart syndrome: A case report and literature review 类卵巢癌合并类癌性心脏综合征1例报告并文献复习
IF 1.3 Q3 OBSTETRICS & GYNECOLOGY Pub Date : 2025-12-01 DOI: 10.1016/j.gore.2025.101997
Minyoung Jang , Lakeisha Mulugeta-Gordon , Joseph Carver , Natalie Tupper , Julie Barbera , Lauren Schwartz , Nawar Latif

Background

Primary ovarian carcinoid tumors are rare neuroendocrine neoplasms comprising <0.1% of ovarian tumors and 1% of all carcinoid tumors. Up to 30% present with carcinoid syndrome, and approximately one-quarter of these develop carcinoid heart syndrome. Unlike gastrointestinal carcinoid tumors, ovarian lesions can cause carcinoid syndrome without hepatic metastases due to direct venous drainage into the systemic circulation.

Case

A 46-year-old woman presented with abdominal discomfort, new onset hypertension, and lower extremity edema. Imaging revealed a 16.8 cm right adnexal mass with mixed solid and cystic components. Echocardiography demonstrated torrential tricuspid regurgitation related to thickened, immobile leaflets, consistent with carcinoid heart syndrome. Serum serotonin and chromogranin A were markedly elevated. She underwent exploratory laparotomy, total abdominal hysterectomy, bilateral salpingo-oophorectomy, right pelvic and para-aortic lymphadenectomy, and omental biopsy. Pathology showed a carcinoid tumor arising in a mature cystic teratoma, confined to the right ovary (FIGO stage IA). Immunohistochemistry confirmed neuroendocrine differentiation (synaptophysin, chromogranin positive). Postoperatively, chromogranin A normalized, serotonin decreased, and proBNP improved. Three months later, she underwent successful bioprosthetic tricuspid valve replacement with ongoing Cardiology follow up.

Discussion

This case illustrates that ovarian carcinoid tumors may present with minimal symptoms despite significant cardiac involvement. Early cardiac evaluation is warranted in suspected carcinoid tumors to identify carcinoid heart syndrome. Overall survival is generally favorable for early stage ovarian carcinoid tumors. Long-term cardiac prognosis is determined by right ventricular function and presence of residual tumor after resection. Management requires individualized and multidisciplinary surgical planning, balancing tumor resection with timely cardiac intervention.
原发性卵巢类癌是一种罕见的神经内分泌肿瘤,占卵巢肿瘤的0.1%,占所有类癌的1%。高达30%的人表现为类癌综合征,其中约四分之一的人发展为类癌心脏综合征。与胃肠道类癌不同,卵巢病变由于静脉直接引流进入体循环,可引起无肝转移的类癌综合征。病例1:46岁女性,腹部不适,新发高血压,下肢水肿。影像显示右侧附件16.8 cm肿块,有实性和囊性混合成分。超声心动图显示剧烈的三尖瓣反流与增厚、不动的小叶有关,符合类癌心脏综合征。血清5 -羟色胺和嗜铬粒蛋白A明显升高。她接受了剖腹探查、全腹子宫切除术、双侧输卵管卵巢切除术、右侧骨盆和主动脉旁淋巴结切除术和大网膜活检。病理显示成熟囊性畸胎瘤的类癌,局限于右侧卵巢(FIGO IA期)。免疫组化证实神经内分泌分化(synaptophysin, chromogranin阳性)。术后,色粒蛋白A正常化,血清素降低,proBNP改善。三个月后,她成功接受了生物假体三尖瓣置换术,并进行了心脏病学随访。本病例表明,尽管严重累及心脏,但卵巢类癌可能表现为轻微症状。对于疑似类癌肿瘤,早期心脏评估是必要的,以确定类癌心脏综合征。早期类卵巢肿瘤的总体生存率通常是有利的。长期心脏预后是由右心室功能和肿瘤切除后是否存在残余肿瘤决定的。治疗需要个体化和多学科的手术计划,平衡肿瘤切除和及时的心脏介入治疗。
{"title":"Ovarian carcinoid tumor with carcinoid heart syndrome: A case report and literature review","authors":"Minyoung Jang ,&nbsp;Lakeisha Mulugeta-Gordon ,&nbsp;Joseph Carver ,&nbsp;Natalie Tupper ,&nbsp;Julie Barbera ,&nbsp;Lauren Schwartz ,&nbsp;Nawar Latif","doi":"10.1016/j.gore.2025.101997","DOIUrl":"10.1016/j.gore.2025.101997","url":null,"abstract":"<div><h3>Background</h3><div>Primary ovarian carcinoid tumors are rare neuroendocrine neoplasms comprising &lt;0.1% of ovarian tumors and 1% of all carcinoid tumors. Up to 30% present with carcinoid syndrome, and approximately one-quarter of these develop carcinoid heart syndrome. Unlike gastrointestinal carcinoid tumors, ovarian lesions can cause carcinoid syndrome without hepatic metastases due to direct venous drainage into the systemic circulation.</div></div><div><h3>Case</h3><div>A 46-year-old woman presented with abdominal discomfort, new onset hypertension, and lower extremity edema. Imaging revealed a 16.8 cm right adnexal mass with mixed solid and cystic components. Echocardiography demonstrated torrential tricuspid regurgitation related to thickened, immobile leaflets, consistent with carcinoid heart syndrome. Serum serotonin and chromogranin A were markedly elevated. She underwent exploratory laparotomy, total abdominal hysterectomy, bilateral salpingo-oophorectomy, right pelvic and para-aortic lymphadenectomy, and omental biopsy. Pathology showed a carcinoid tumor arising in a mature cystic teratoma, confined to the right ovary (FIGO stage IA). Immunohistochemistry confirmed neuroendocrine differentiation (synaptophysin, chromogranin positive). Postoperatively, chromogranin A normalized, serotonin decreased, and proBNP improved. Three months later, she underwent successful bioprosthetic tricuspid valve replacement with ongoing Cardiology follow up.</div></div><div><h3>Discussion</h3><div>This case illustrates that ovarian carcinoid tumors may present with minimal symptoms despite significant cardiac involvement. Early cardiac evaluation is warranted in suspected carcinoid tumors to identify carcinoid heart syndrome. Overall survival is generally favorable for early stage ovarian carcinoid tumors. Long-term cardiac prognosis is determined by right ventricular function and presence of residual tumor after resection. Management requires individualized and multidisciplinary surgical planning, balancing tumor resection with timely cardiac intervention.</div></div>","PeriodicalId":12873,"journal":{"name":"Gynecologic Oncology Reports","volume":"62 ","pages":"Article 101997"},"PeriodicalIF":1.3,"publicationDate":"2025-12-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145620214","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
A fast-progressing orthotopic ovarian cancer model reveals synergistic antitumor effects of AXL-targeting nanobodies and Olaparib 一个快速进展的原位卵巢癌模型揭示了靶向axl的纳米体和奥拉帕尼的协同抗肿瘤作用
IF 1.3 Q3 OBSTETRICS & GYNECOLOGY Pub Date : 2025-12-01 DOI: 10.1016/j.gore.2025.101998
Aarushi A. Caro , Alícia Gordún Peiró , Eva Hadadi , Yani Berckmans , Yvon Elkrim , Ayla Debraekeleer , Gisèle Mateu Cabrera , Kim De Veirman , An Coosemans , Damya Laoui

Objective

Ovarian cancer (OC) is the deadliest gynecological malignancy, with patients experiencing late diagnosis and high recurrence. AXL is highly expressed in OC and correlates with worse prognosis. We aim to evaluate the use of AXL-targeting nanobodies as a novel therapeutic modality to inhibit the AXL-GAS6 axis in OC.

Methods

ID8-fLuc cells were in vivo passaged three times to obtain the fast-progressing P3 ID8 Thy1.1 cell line. The expression of AXL in ID8 lines was assessed by flow cytometry and immunofluorescence, while serum GAS6 levels in tumor-bearing and naïve mice were measured by ELISA. Using in silico analyses, we correlated AXL and GAS6 expression with OC patient outcomes and assessed AXL expression in different tissues and patient cohorts. Orthotopic ovarian tumors were processed to a single cell suspension and treated in vitro with the nanobody (AXL-Fc), a PARP inhibitor (Olaparib), or controls (PBS or DMSO) for 24 h or 72 h, followed by flow cytometry to assess stages of cell death and cancer cell proliferation.

Results

In vivo passaging of ID8-fLuc cells resulted in a faster-progressing P3 ID8 Thy1.1 cell line that recapitulates stages I-III observed in OC patients. In OC patients, AXL and GAS6 genes are highly expressed in primary and metastatic tumors, and enriched in platinum-resistant patients. AXL-Fc induces necrosis in OC cells from orthotopic ovarian tumors. AXL-Fc synergizes with Olaparib, resulting in decreased OC cell proliferation.

Conclusions

Leveraging the synergistic effects of AXL-Fc with Olaparib, we propose a new AXL-targeting treatment approach for OC that warrants further investigation.
目的卵巢癌是最致命的妇科恶性肿瘤,诊断晚,复发率高。AXL在OC中高表达,预后较差。我们的目的是评估使用靶向axl的纳米体作为一种新的治疗方式来抑制OC中的AXL-GAS6轴。方法sid8 - fluc细胞在体内传代3次,获得快速进展的P3 ID8 Thy1.1细胞系。采用流式细胞术和免疫荧光法检测ID8细胞系中AXL的表达,ELISA法检测荷瘤小鼠和naïve小鼠血清中GAS6的表达。通过计算机分析,我们将AXL和GAS6的表达与OC患者的预后相关联,并评估了AXL在不同组织和患者队列中的表达。将原位卵巢肿瘤处理成单细胞悬液,用纳米体(AXL-Fc)、PARP抑制剂(Olaparib)或对照组(PBS或DMSO)在体外处理24小时或72小时,然后用流式细胞术评估细胞死亡和癌细胞增殖的阶段。结果ID8- fluc细胞的体内传代导致P3 - ID8 Thy1.1细胞系进展更快,重现了在OC患者中观察到的I-III期。在OC患者中,AXL和GAS6基因在原发和转移性肿瘤中高表达,在铂耐药患者中富集。AXL-Fc诱导原位卵巢肿瘤OC细胞坏死。AXL-Fc与奥拉帕尼协同作用,导致OC细胞增殖下降。结论利用AXL-Fc与奥拉帕尼的协同作用,我们提出了一种新的靶向axl治疗OC的方法,值得进一步研究。
{"title":"A fast-progressing orthotopic ovarian cancer model reveals synergistic antitumor effects of AXL-targeting nanobodies and Olaparib","authors":"Aarushi A. Caro ,&nbsp;Alícia Gordún Peiró ,&nbsp;Eva Hadadi ,&nbsp;Yani Berckmans ,&nbsp;Yvon Elkrim ,&nbsp;Ayla Debraekeleer ,&nbsp;Gisèle Mateu Cabrera ,&nbsp;Kim De Veirman ,&nbsp;An Coosemans ,&nbsp;Damya Laoui","doi":"10.1016/j.gore.2025.101998","DOIUrl":"10.1016/j.gore.2025.101998","url":null,"abstract":"<div><h3>Objective</h3><div>Ovarian cancer (OC) is the deadliest gynecological malignancy, with patients experiencing late diagnosis and high recurrence. AXL is highly expressed in OC and correlates with worse prognosis. We aim to evaluate the use of AXL-targeting nanobodies as a novel therapeutic modality to inhibit the AXL-GAS6 axis in OC.</div></div><div><h3>Methods</h3><div>ID8-fLuc cells were <em>in vivo</em> passaged three times to obtain the fast-progressing P3 ID8 Thy1.1 cell line. The expression of AXL in ID8 lines was assessed by flow cytometry and immunofluorescence, while serum GAS6 levels in tumor-bearing and naïve mice were measured by ELISA. Using <em>in silico</em> analyses, we correlated AXL and GAS6 expression with OC patient outcomes and assessed AXL expression in different tissues and patient cohorts. Orthotopic ovarian tumors were processed to a single cell suspension and treated <em>in vitro</em> with the nanobody (AXL-Fc), a PARP inhibitor (Olaparib), or controls (PBS or DMSO) for 24 h or 72 h, followed by flow cytometry to assess stages of cell death and cancer cell proliferation.</div></div><div><h3>Results</h3><div><em>In vivo</em> passaging of ID8-fLuc cells resulted in a faster-progressing P3 ID8 Thy1.1 cell line that recapitulates stages I-III observed in OC patients. In OC patients, <em>AXL</em> and <em>GAS6</em> genes are highly expressed in primary and metastatic tumors, and enriched in platinum-resistant patients. AXL-Fc induces necrosis in OC cells from orthotopic ovarian tumors. AXL-Fc synergizes with Olaparib, resulting in decreased OC cell proliferation.</div></div><div><h3>Conclusions</h3><div>Leveraging the synergistic effects of AXL-Fc with Olaparib, we propose a new AXL-targeting treatment approach for OC that warrants further investigation.</div></div>","PeriodicalId":12873,"journal":{"name":"Gynecologic Oncology Reports","volume":"62 ","pages":"Article 101998"},"PeriodicalIF":1.3,"publicationDate":"2025-12-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145620213","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Somatically derived ovarian yolk sac tumor in a postmenopausal woman: A case report of durable remission with personalized chemotherapy 绝经后女性体内衍生性卵巢卵黄囊肿瘤:个体化化疗持久缓解一例报告
IF 1.3 Q3 OBSTETRICS & GYNECOLOGY Pub Date : 2025-12-01 DOI: 10.1016/j.gore.2025.101989
Vinita Popat , Adam Rucker , Joseph W. Carlson , Aimee Keegan , Lorna Rodriguez-Rodriguez

Background

Somatically derived ovarian yolk sac tumors (SD-YSTs) in postmenopausal women are rare and typically present with advanced-stage disease, often carrying a poor prognosis.

Case

We describe a 74-year-old woman with stage IIIB SD-YST who underwent complete surgical resection followed by cisplatin-etoposide chemotherapy, with bleomycin omitted due to frailty. Chemotherapy scheduling was modified in real time according to alpha-fetoprotein (AFP) kinetics, with treatment intervals shortened from 21 to 14 days after AFP levels rose between early cycles. Despite dose reductions due to thrombocytopenia, AFP levels normalized after cycle 3, and treatment was discontinued after cycle 4.

Outcome

The patient has remained disease-free for over five years with ongoing surveillance.

Conclusion

This case underscores the potential for long-term remission in SD-YST with platinum-based therapy, even when standard regimens require modification for older or frail patients. It highlights the value of biomarker-guided treatment adjustments to optimize chemotherapy timing in rare ovarian malignancies.
卵巢卵黄囊肿瘤(SD-YSTs)在绝经后妇女中很少见,通常出现在晚期,通常预后较差。病例:我们描述了一位74岁的IIIB期SD-YST女性患者,她接受了完整的手术切除,随后进行了顺铂-依托泊苷化疗,由于虚弱而省略了博来霉素。化疗计划根据甲胎蛋白(AFP)动力学实时修改,治疗间隔从早期周期甲胎蛋白水平升高后的21天缩短到14天。尽管由于血小板减少导致剂量减少,但AFP水平在第3周期后恢复正常,并在第4周期后停止治疗。结果:患者在持续监测的情况下无病已超过5年。结论:该病例强调了铂基治疗SD-YST长期缓解的潜力,即使标准方案需要对老年或体弱患者进行修改。它强调了生物标志物引导的治疗调整的价值,以优化罕见卵巢恶性肿瘤的化疗时机。
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引用次数: 0
Etoposide combined with anlotinib and sintilimab successfully achieved long term progression-free survival in platinum-refractory ovarian cancer: A case report 依托泊苷联合安洛替尼和辛替单抗治疗铂难治性卵巢癌成功实现长期无进展生存:1例报告
IF 1.3 Q3 OBSTETRICS & GYNECOLOGY Pub Date : 2025-12-01 DOI: 10.1016/j.gore.2025.101999
Luying Zhang, Yuxin Wang, Cang Qiu, Yang Li, Kelei Zhao, Xiaohan Yuan, Yanting Liu, Ping Lu, Min Zhang

Background

Platinum-refractory ovarian cancer (PROC) is extremely malignant and aggressive. Patients typically have a diminished quality of life and limited survival, with an overall survival (OS) of less than one year. Until early 2024, bevacizumab remains the only targeted agent approved in China for the treatment of PROC. The standard regimen for PROC in China is non-platinum single-agent chemotherapy, or chemotherapy combined with bevacizumab. However, these therapies have not achieved satisfactory clinical outcomes.

Case presentation

A 63-year-old woman presented with an incidentally detected left neck mass. Ultrasound-guided biopsy revealed high-grade serous ovarian carcinoma. She received first-line bevacizumab plus platinum-based chemotherapy but showed disease progression after two cycles, consistent with platinum-refractory ovarian cancer. After similarly failing second-line gemcitabine therapy (2 cycles), she achieved complete response (CR) and maintained progression-free survival (PFS) for 23.9 months with third-line etoposide combined with anlotinib and sintilimab.

Conclusion

This case demonstrates that the combination of etoposide, anlotinib, and sintilimab can induce sustained CR in platinum-refractory ovarian cancer, achieving remarkable 23.9-month PFS. The synergistic activity of this regimen successfully reversed multidrug resistance and may redefine third-line therapeutic strategies for this challenging population.
背景铂难治性卵巢癌(PROC)是一种极具恶性和侵袭性的肿瘤。患者通常生活质量下降,生存期有限,总生存期(OS)不到一年。直到2024年初,贝伐单抗仍是中国唯一批准用于治疗PROC的靶向药物,中国PROC的标准方案是非铂单药化疗,或化疗联合贝伐单抗。然而,这些疗法并没有取得令人满意的临床效果。病例表现一名63岁女性,偶然发现左颈部肿块。超声引导下活检显示高级别浆液性卵巢癌。她接受了一线贝伐单抗加铂基化疗,但在两个周期后出现疾病进展,与铂难治性卵巢癌一致。在二线吉西他滨治疗同样失败(2个周期)后,她获得了完全缓解(CR),并在三线依托泊苷联合安洛替尼和辛替单抗的情况下维持了23.9个月的无进展生存期(PFS)。结论依托泊苷、安洛替尼、辛替单抗联合治疗铂难治性卵巢癌可诱导持续CR,取得显著的23.9个月PFS。该方案的协同作用成功逆转了多药耐药,并可能重新定义针对这一具有挑战性人群的三线治疗策略。
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引用次数: 0
Considering homologous recombination deficiency status in HIPEC: could molecular testing someday drive “targeted” surgery? 考虑到HIPEC中同源重组缺陷的状况:分子检测有朝一日能推动“靶向”手术吗?
IF 1.3 Q3 OBSTETRICS & GYNECOLOGY Pub Date : 2025-12-01 DOI: 10.1016/j.gore.2025.102001
Sarah P. Huepenbecker , Katherine C. Fuh
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引用次数: 0
期刊
Gynecologic Oncology Reports
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