BMC Pregnancy and Childbirth

The influence of uterine fibroids on adverse outcomes in pregnant women: a meta-analysis

Abstract Objective The objective of the meta-analysis was to determine the influence of uterine fibroids on adverse outcomes, with specific emphasis on multiple or large (≥ 5 cm in diameter) fibroids. Materials and methods We searched PubMed, Embase, Web of Science, ClinicalTrials.gov, China National Knowledge Infrastructure (CNKI), and SinoMed databases for eligible studies that investigated the influence of uterine fibroids on adverse outcomes in pregnancy. The pooled risk ratio (RR) of the variables was estimated with fixed effect or random effect models. Results Twenty-four studies with 237 509 participants were included. The pooled results showed that fibroids elevated the risk of adverse outcomes, including preterm birth, cesarean delivery, placenta previa, miscarriage, preterm premature rupture of membranes (PPROM), placental abruption, postpartum hemorrhage (PPH), fetal distress, malposition, intrauterine fetal death, low birth weight, breech presentation, and preeclampsia. However, after adjusting for the potential factors, negative effects were only seen for preterm birth, cesarean delivery, placenta previa, placental abruption, PPH, intrauterine fetal death, breech presentation, and preeclampsia. Subgroup analysis showed an association between larger fibroids and significantly elevated risks of breech presentation, PPH, and placenta previa in comparison with small fibroids. Multiple fibroids did not increase the risk of breech presentation, placental abruption, cesarean delivery, PPH, placenta previa, PPROM, preterm birth, and intrauterine growth restriction. Meta-regression analyses indicated that maternal age only affected the relationship between uterine fibroids and preterm birth, and BMI influenced the relationship between uterine fibroids and intrauterine fetal death. Other potential confounding factors had no impact on malposition, fetal distress, PPROM, miscarriage, placenta previa, placental abruption, and PPH. Conclusion The presence of uterine fibroids poses increased risks of adverse pregnancy and obstetric outcomes. Fibroid size influenced the risk of breech presentation, PPH, and placenta previa, while fibroid numbers had no impact on the risk of these outcomes.

A meta-analysis of treatment for early-stage cervical cancer: open versus minimally invasive radical trachelectomy

Abstract Background In previous systematic reviews, meta-analysis was lacking, resulting in the statistical difference between the data of different surgeries being impossible to judge. This meta-analysis aims to contrast the fertility results and cancer outcomes between open and minimally invasive surgery. Method We systematically searched databases including PubMed, Embase, Cochrane, and Scopus to collect studies that included open and minimally invasive radical trachelectomy. A random-effect model calculated the weighted average difference of each primary outcome via Review Manager V.5.4. Result Eight studies (1369 patients) were incorporated into our study. For fertility results, the Open group excels MIS group in pregnancies-Third trimester delivery [OR = 2.68; 95% CI (1.29, 5.59); P  = 0.008]. Nevertheless, there is no statistical difference in clinical pregnancy, miscarriage, and second-trimester rate. Concerning cancer outcomes, no difference was detected in the overall survival [OR = 1.56; 95% CI (0.70, 3.45); P  = 0.27] and recurrence [OR = 0.63; 95% CI (0.35, 1.12); P  = 0.12]. Concerning surgery-related outcomes, the comprehensive effects revealed that the estimated blood loss of the Open group was higher than that of the MIS group[MD = 139.40; 95% CI (79.05, 199.75); P  < 0.0001]. However, there was no difference between the postoperative complication rate in the two groups [OR = 1.52; 95% CI (0.89, 2.60); P  = 0.12]. Conclusion This meta-analysis suggested that the fertility result of the Open group may be better than the MIS group, while the MIS group has better surgery-related outcomes. Owing to the poor cases of our study, a more robust conclusion requires more relevant articles in the future. Systematic review registration PROSPERO CRD42022352999.

Yolk sac tumor and dysgerminoma in the left gonad following gonadoblastoma in the right gonad in a 46,XY DSD with a novel SRY missense mutation: a case report

Abstract Background Approximately 10–15% of 46,XY disorders of sex development (DSDs) have an SRY mutation residing in the high mobility group (HMG) domain. Here, we present a case of 46,XY DSD caused by a novel missense mutation in the HMG region of SRY rapidly progressing to germ cell tumors (GCTs). Case presentation An adolescent female (15 years old) exhibiting primary amenorrhea was later diagnosed as a 46,XY female with bilateral gonadal dysplasia on the basis of peripheral lymphocyte karyotype 46,XY and a novel missense mutation in SRY (c.281 T > G, p.L94R). The novel missense mutation (c.281 T > G, p.L94R) and its adjacent region were conserved. Protein structure analysis showed that the mutant site was located in the middle of the HMG domain, and the mutant protein had a diminished ability to bind to DNA. Imaging examination revealed an adolescent female with a naive uterus. Laparoscopy and initial pathological examination revealed left gonadal dysplasia and right gonadal dysplasia with gonadoblastoma (GB). Right gonadectomy by laparoscopy was performed upon consent from the patient’s parents. Less than 1 year postoperatively, the left gonadal gland deteriorated as observed by the findings of a mass in the left adnexal region by pelvic MRI and serum AFP > 1000 ng/ml by serological tests, and then total hysterectomy and adnexal and left gonadectomy by laparoscopy were performed. The GCT stage was classified as stage Ic according to FIGO. At this time, pathologic examination showed that the left gonad had progressed to yolk sac tumor and dysgerminoma. The patient underwent chemotherapy post-operatively but developed type III myelosuppression and tumor recurrence several months later. Conclusions The patient initially presented with right gonadoblastoma but chose only right gonadectomy by laparoscopy to preserve the female sex characteristics, which resulted in rapid deterioration of the left gonad and poor treatment outcomes. This case demonstrates the importance of early genetic diagnosis and treatment of 46,XY female DSD.

Intraplacental choriocarcinoma in a term pregnancy with massive fetomaternal hemorrhage: a case report

Intraplacental choriocarcinoma (IC) is a rare type of gestational choriocarcinoma that occurs in the placenta, with only a small number of cases reported. The symptoms of IC are nonspecific, and the diagnosis is often incidental, typically made during routine placental pathological examination. We report a case of IC in a 30-year-old Chinese woman presenting with intrauterine fetal distress and fetomaternal hemorrhage. The patient was admitted to our hospital at 40 weeks of gestation due to decreased fetal movements. An emergency cesarean section was performed, delivering a male infant with anemic appearance. Laboratory tests confirmed fetomaternal hemorrhage syndrome, while microscopic examination of the placenta revealed intraplacental choriocarcinoma. Targeted placental examination is advisable for patients with pregnancy complications. We hope this case raises awareness of this potential scenario, enabling clinicians to identify similar cases more readily in the future.

Dysfunctional labor and hemoperitoneum secondary to an incidentally discovered dysgerminoma: a case report

Abstract Background Ovarian dysgerminoma, a subtype of malignant germ cell tumor (GCT), is a rare ovarian neoplasm that is infrequently found in the gravid patient. When dysgerminomas do occur in pregnancy, the rapidly growing tumors can have a heterogeneous presentation and lead to peripartum complications and morbidity. Due to the rarity of this condition, diagnostic and therapeutic strategies are not well described in the literature. Case presentation A healthy multigravida with an uncomplicated antenatal history presented for elective induction of labor. She had a protracted labor course, persistently abnormal cervical examinations, and eventually developed a worsening Category II tracing that prompted cesarean birth. Intraoperatively, a 26 cm pelvic mass later identified as a Stage IA dysgerminoma was discovered along with a massive hemoperitoneum. The mass was successfully resected, and the patient remains without recurrence 6 months postoperatively. Conclusion Although rare and generally indolent, dysgerminomas can grow rapidly and cause mechanical obstruction of labor and other complications in pregnancy. Pelvic masses, including malignant neoplasms, should be included in as part of a broad differential diagnosis when evaluating even routine intrapartum complications such as abnormal labor progression. Additionally, we demonstrate that adnexal masses can be a source of life-threatening intraabdominal hemorrhage.

Pregnancies after vaginal radical trachelectomy (RT) in patients with early invasive uterine cervical cancer: results from a single institute

Abstract Background Radical tracheletomy (RT) with pelvic lymphadenectomy has become an option for young patients with early invasive uterine cervical cancer who desire to maintain their fertility. However, this operative method entails a high risk for the following pregnancy due to its radicality. Methods We have performed vaginal RT for 71 patients and have experienced 28 pregnancies in 21 patients. They were followed up carefully according to the follow-up methods we reported previously. Their pregnancy courses and prognoses after the pregnancy were retrospectively reviewed. Results All the vaginal RTs were performed safely without serious complications, including 6 patients who underwent the operation during pregnancy. The median time to be pregnant after RT was 29.5 months. 13 patients (46%) became pregnant without artificial insemination by husband or assisted reproductive technology. Cesarean section was performed for all of them. The median time of pregnancy was 34 weeks, and emergent cesarean section was performed for 7 pregnancies (25%). The median birth weight was 2156 g. Four patients had trouble with cervical cerclage, and they suffered from sudden premature preterm rupture of the membrane (pPROM) during the second trimester of pregnancy. We underwent transabdominal cerclage (TAC) for all of them and careful management for the prevention of uterine infection was performed. One patient had a recurrence of cancer during pregnancy. Conclusions Both the obstetrical prognosis and oncological prognosis after vaginal RT have become favorable for pregnant patients after vaginal RT.

A case of successful maintained pregnancy after neoadjuvant chemotherapy plus radical surgery for stage IB3 cervical cancer diagnosed at 13 weeks

Abstract Background Cervical cancer during pregnancy is rare. The management for stage IB3 during pregnancy remains unclear and challenging. We report a successful preserved pregnancy in a stage IB3 patient who was treated with neoadjuvant chemotherapy (NACT) plus radical surgery. Case presentation A 36-year-old pregnant woman was diagnosed with a 5-cm-diameter stage IB3 squamous cell carcinoma of the uterine cervix at 13 gestational weeks. The patient received 5 courses of systemic chemotherapy with carboplatin and paclitaxel every 3 weeks, followed by caesarean section and radical hysterectomy. Both the mother and infant are in good general condition. Conclusion This case suggests that NACT plus radical surgery could be one method to maintain pregnancy in stage IB3 cervical cancer diagnosed as early as 13 gestational weeks.

Spontaneous regression of a giant uterine leiomyoma after delivery: a case report and literature review

Abstract Background Uterine leiomyomas are hormone-dependent benign tumors and often begin to shrink after menopause due to the reduction in ovarian steroids. The influence of pregnancy on uterine leiomyomas size remains unclear. Here, we present a case of spontaneous regression of a giant uterine leiomyoma after delivery. Case presentation A 40-year-old woman presented with multiple uterine leiomyomas, one of which is a giant uterine leiomyomas (approximately 8 cm in diameter) that gradually shrinked after delivery. At over two months postpartum, the large myometrial leiomyoma had transformed into a submucosal leiomyoma, and over 3 years postpartum, both the submucosal leiomyoma and multiple intramural leiomyomas completely regressed. Conclusion Spontaneous regression of a giant uterine leiomyom is rare after delivery. Considering uterine leiomyoma regression until over 3 year postpartum,we need to observe the regression of uterine fibroid for a longer time postpartum in the absence of fibroid related complications. In addition, it will provide new insights for treatment options of uterine leiomyomas in the future.

Endometrioid carcinomas with sex cord-like formations and hyalinization: spontaneous pregnancy after conservative treatment

AbstractEndometrioid carcinoma with sex cord-like formations and hyalinization of the uterine corpus, or corded and hyalinized endometrioid adenocarcinoma (CHEC), is a rare morphological variant of endometrioid carcinoma, for which there is limited literature and few cases reports. Most researchers tend to consider CHEC as a low-grade cancer with a favorable prognosis. Full-staging surgery is the primary choice for this disease, and no case of CHEC has been previously reported to be treated conservatively. Here, we present the following case to explore the possibility of fertility-preserving treatment for young women with CHEC. A 23-year-old nulliparous patient diagnosed with presumed stage IA CHEC received fertility-sparing treatment at the Obstetrics and Gynecology Hospital of Fudan University and got a complete response (CR) after 10 months of conservative treatment. The patient subsequently became pregnant spontaneously, successfully conceived, and gave birth to a healthy male neonate without any sign of recurrence during 37 months follow-up after CR. The patient’s postpartum follow-up is continuing. Presently, CHEC is not included in the fertility-sparing field of any available guidelines. This case indicates that fertility-sparing treatment may be an option for highly selected patients with CHEC. Continuous follow-up remains mandatory to observe long-term outcomes.

Successful delivery after in vitro fertilization-embryo transfer in a woman with metachronous primary cancer of ovary and endometrium: a case report

Abstract Background The appearance of malignancies at various times in the same individual, excluding metastases of the initial primary cancer, is termed multiple primary cancers. Double primary gynecological cancers cause inevitable damage to female reproductive function, and the preservation of fertility in such patients remains a challenging issue as relatively few cases have been reported. This case report provides management options for dual primary ovarian and endometrial cancers, including the choice of ovulation induction protocols, considerations during pregnancy and parturition, with the aim of providing assistance to clinicians. Case presentation We report a case of a 39-year-old woman with primary infertility and a medical history of right-sided ovarian mucinous borderline tumor with intraepithelial carcinoma, left-sided ovarian mucinous cystadenoma and endometrial cancer, who successfully conceived with in vitro fertilization-embryo transfer (IVF-ET) after three different ovulation induction protocols. During her pregnancy, she was complicated by central placenta praevia with placental implantation and eventually delivered a healthy female infant by caesarean section at 33 gestational weeks. Conclusions For patients with double primary gynecological cancers who have an intense desire for fertility, the most appropriate oncological treatment should be applied according to the patient’s individual situation, and fertility preservation should be performed promptly. Ovulation induction protocol should be individualized and deliberate, with the aim of ensuring that the patient’s hormone levels do not precipitate a recurrence of the primary disease during induction of ovulation and maximizing fertility outcomes. In addition, the risk of postpartum hemorrhage due to placental factors cannot be neglected in such patients.

Recurrent endometrial atypical hyperplasia diagnosed by pathological examination of the placenta from a live birth: a case report

Abstract Background Pregnancy complicated with endometrial atypical hyperplasia, which is often observed during early pregnancy, is extremely rare. Case presentation The patient was a 30-year-old woman who had premature delivery at 30+ 1 weeks gestation, and endometrial atypical hyperplasia was discovered by placental examination. Conclusions For patients who undergo fertility-sparing treatment for endometrial atypical hyperplasia, the evaluation of the decidua via the placental pathological examination is particularly important. These examinations make a great clinical contribution to the early detection and diagnosis of endometrial atypical hyperplasia.

Early ultrasound diagnosis and management of intramural pregnancy: a case series report utilizing the FIGO leiomyoma classification system and 3D sonographic mapping

Intramural pregnancy, a rare ectopic pregnancy with the gestational sac implanted within the myometrium, poses significant diagnostic and therapeutic challenges. Current diagnostic criteria lack anatomical precision, leading to delayed management and high morbidity. This study proposes a novel diagnostic framework integrating the International Federation of Gynecology and Obstetrics (FIGO) leiomyoma classification system and advanced 3D transvaginal ultrasound, enabling anatomical risk stratification and individualized management. We retrospectively analyzed four intramural pregnancies, assessing clinical history, laboratory results, 2D/3D sonographic findings, treatment methods and outcomes. All cases underwent comprehensive evaluation using high-resolution transvaginal 2D/3D ultrasound (GE Voluson E8 or S8 systems with 5-6.5 MHz transvaginal probes). Lesions were systematically mapped according to the FIGO leiomyoma classification system, with particular attention to their spatial relationship with the uterine cavity and surrounding myometrium. Treatment strategies were tailored based on lesion characteristics, β-hCG levels, and fertility goals. Two distinct sonographic patterns were identified: gestational sac-type (Cases 1-2) and heterogeneous mass-type (Cases 3-4). The FIGO classification system (Types 2-5) effectively predicted the depth of myometrial invasion and guided therapeutic decisions. Type 3 (Case 1): Medical management with methotrexate (MTX) achieved resolution. Type 2-5 (Case 2): Ultrasound-guided fetal reduction preserved intrauterine pregnancy to term. Type 4 and Type 5 (Cases 3-4): Laparoscopic excision prevented uterine rupture, with lesions demonstrating significant myometrial thinning or serosal protrusion. High risk factors and key sonographic features are summarized, and diagnostic criteria are proposed. The integration of FIGO-based anatomical staging with advanced 3D sonographic evaluation provides a robust framework for early diagnosis and risk-stratified management of intramural pregnancy. This innovative approach significantly enhances diagnostic accuracy, facilitates appropriate treatment selection, and optimizes fertility preservation outcomes.

Fetal biometric and Doppler measurements following abdominal radical trachelectomy in the second trimester of the pregnancy

Abstract Background Our previous study demonstrated the safety and effectiveness of abdominal radical trachelectomy during pregnancy but did not focus on the fetus. This study aimed to clarify the influence of abdominal radical trachelectomy performed during pregnancy on the fetus. Methods Eight cervical cancer patients who underwent abdominal radical trachelectomy at our hospital between February 2013 and August 2020 were enrolled in this study. To assess the peri- and postoperative influence on the fetus, we performed fetal heart monitoring at 30-min intervals during abdominal radical trachelectomy and calculated the estimated fetal body weight and resistance indexes of the middle cerebral artery and umbilical artery from postsurgery until delivery. Results Four out of eight patients had preterm birth due to chorioamnionitis in one case and consideration of the recurrent risk of cervical cancer in three cases. Fetal heart monitoring during abdominal radical trachelectomy revealed deceleration just once in one case but no abnormal findings in the other cases. In all cases, the fetal growth after abdominal radical trachelectomy was normal until delivery. No abnormal Doppler findings were detected in the middle cerebral artery or umbilical artery. Conclusion Our findings clarified that abdominal radical trachelectomy performed for the treatment of early-stage cervical cancer during pregnancy has no obvious influence on fetal growth. Next, it is necessary to evaluate the growth and development of children delivered from mothers who have undergone abdominal radical trachelectomy during pregnancy.

Cervical neuroendocrine carcinoma in the third trimester: a rare case report and literature review

Abstract Background The incidence of cervical neuroendocrine carcinoma (CNECC) during pregnancy is extremely rare. Insufficient awareness of gynecological tumor screening, as well as atypical and insidious initial clinical symptoms, may result in diagnostic delays and misdiagnosis. There is no standardized treatment for cervical cancer in pregnancy. Herein, we present a case of cervical neuroendocrine carcinoma diagnosed in the third trimester of pregnancy. Case presentation A 26-year-old female at 30 1/7 weeks of gestation presented with lower back and sacroiliac joint pain, abdominal distension, and lower limb dyskinesia. A pelvic examination revealed a large fungating gray mass that encompassed the entire cervix, with cervical contact bleeding testing positive. Imaging studies showed a significant cervical mass, diffuse liver changes, and metastasis to multiple sites. Biopsy results revealed poorly differentiated cervical carcinoma exhibiting high-grade neuroendocrine features, consistent with a diagnosis of large cell neuroendocrine carcinoma. The patient was diagnosed with stage IVB CNECC with HPV18 (+), and due to the gestational age of the fetus and her deteriorating condition, she underwent cesarean section delivery after receiving fetal lung maturation therapy. Following surgery, eight cycles of neoadjuvant chemotherapy were applied. Unfortunately, she succumbed to multiple tumor metastases six months post-treatment. Despite this tragic outcome, the infant was discharged in a healthy condition. Conclusions CNECC during pregnancy, particularly the large-cell type, is an ultra-rare condition with poor prognosis. This case highlights the importance of individualized treatment approach and the need for better screening, early detection, and treatment. Given the rarity of the disease, further research is warranted to determine the prognostic factors and develop effective treatment strategies for this ultra-rare and aggressive malignancy.

Ovarian ependymoma presenting in pregnancy: a case report and literature review

Abstract Background Ovarian ependymoma is a rare malignancy. Because of the extreme rarity, certain features of the neoplasm, including its clinical behaviour and optimal therapy, are incompletely characterized. Case presentation A 32-year-old pregnant woman at term presented with a left ovarian neoplasm that occurred in the early stage of pregnancy. She underwent left adnexectomy during the caesarean section, and the neoplasm was histologically and immunohistochemically identified to be ovarian ependymoma. Immunohistochemical staining with oestrogen receptors and progesterone receptors showed strong positive staining. According to reports in the literature, the pathological type of ovarian ependymoma in our patient was the extra-axial type. Interestingly, the foetus was also found to have bilateral ependymal cysts during pregnancy. The patient received no further surgical treatment or adjuvant therapy. She and her 14-month-old baby both have no evidence of disease at present. The follow-up of both mother and child is still continuing. Conclusions The case presented here illustrates high levels of oestrogen during pregnancy may stimulate viable malignant ependymal cells to proliferate. Hence, young women with extra-axial-type ependymomas may not be suitable for fertility preservation. Moreover, hormone-based therapies can be a potentially effective treatment for women with extra-axial ependymomas.

Struma ovarii and peritoneal strumosis during pregnancy

Abstract Background Struma ovarii is a special type of ovarian dermoid cyst and accounts for approximately 2–3 % of all dermoid tumours. Benign struma ovarii may manifest as distant metastasis, called peritoneal strumosis, which makes it biologically similar to malignancy, and has been reported in limited cases but never discovered during pregnancy. Case presentation We report a patient with a history of right struma ovarii cystectomy. During pregnancy, pelvic masses with non-specific clinical presentation were found again. During the caesarean section, contralateral struma ovarii with dissemination of nodules in the peritoneal cavity was found, and pathology revealed that the masses were thyroid follicle ovarian goitres. Discussion and conclusions Recurrent benign struma ovarii with extraovarian dissemination is a rare aggressive clinical manifestation different from malignancy. It is emphasized that adequate assessment and complete resection of suspicious masses are of great importance.

Clinical predictors of residual disease in hysterectomy following a loop electrosurgical excision procedure for cervical intraepithelial neoplasia grade 3

Abstract Objective To investigate the predictors of residual disease in a hysterectomy following a loop electrosurgical excision procedure (LEEP) for cervical intraepithelial neoplasia (CIN) 3. Methods This retrospective study identified 421 patients with histologically confirmed CIN 3 who underwent LEEP and subsequently had a hysterectomy within 6 months. The clinical data included age, parity, type of transformation zone, cytology results, human papillomavirus (HPV) genotype test, endocervical curettage (ECC), and pathological data of LEEP and hysterectomy were obtained from the medical records. A logistic regression model was used to analyze the relationship between the variables and the risk of residual disease in the hysterectomy samples. Results 186 (44.18%) patients had residual disease in the hysterectomy specimens. The predictive markers of residual disease following LEEP included positive ECC, positive margin of the samples from LEEP, type II or III transformation zone, HPV16 and HPV18 infection, and other high-risk HPV. HPV-18 positivity (OR, 7.13; 95% CI, 3.49 to 14.56; p < 0.001) and type III transformation zone (OR, 6.37; 95% CI, 2.91 to 13.94; p < 0.001) were the most indicative of residual disease following LEEP. Conclusion Positive high-risk HPV, particularly HPV18, positive ECC, the positive margin of specimens from LEEP, and type II or III transformation zone were reliable prognostic markers of residual disease following a LEEP for CIN 3.

Outcomes of in vitro fertilization for women with thyroid cancer: a case report

Magnitude and predictors of gestational trophoblastic disease in East Africa: a systematic review and meta-analysis

Gestational Trophoblastic Disease (GTD) encompasses a spectrum of placenta-related disorders with significant health risks. Despite the varying reported prevalence rates in East Africa, no comprehensive meta-analysis has been conducted to synthesize the overall burden and predictors of GTD in this region. Following the PRISMA guidelines, we conducted a systematic review and meta-analysis of studies from PubMed, Cochrane Library, and Google Scholar (search period: 2000-2024). We assessed heterogeneity (I Eleven studies were included, revealing a pooled GTD magnitude of 10.5% (95% CI: 8.8-12.3; I Gestational trophoblastic disease remains a significant public health burden in East Africa, where delayed healthcare access contributes to worsened outcomes. Women with a history of gestational trophoblastic disease or high parity constitute high-risk populations that require targeted screening programs and early intervention strategies to improve their detection and management.

Multidisciplinary, whole-process management of a patient with recurrent serous borderline ovarian tumor from embryo cryopreservation before re-fertility-sparing surgery to successful delivery: a case report

For patients with recurrent serous borderline ovarian tumors (BOT) who have undergone unilateral salpingo-oophorectomy, there is a risk of reduced or even lost ovarian reserve after the second surgery; therefore, fertility preservation (FP) prior to re-operation in patients of childbearing age is challenging, and has attracted increasing attention. Here, we discuss the multidisciplinary whole-process management of a patient with recurrent serous BOT, from embryo cryopreservation (EC) before re-fertility-sparing surgery (re-FSS) to successful delivery. We describe the treatment of a 28-year-old married, nulliparous female with recurrent serous BOT who requested FP. The patient underwent right salpingo-oophorectomy in July 2020 for serous BOT. In March 2023, B-ultrasound indicated BOT recurrence, and she underwent a second operation. After a multidisciplinary discussion and information on the risks, the patient strongly requested EC. We used letrozole (LE) combined with an antagonist for ovarian stimulation; 16 eggs were obtained, 15 eggs were MII, four embryos on the 3rd day and one blastocyst was formed and cryopreserved. One month later, laparoscopic cystectomy was performed, and pathological examination revealed serous BOT. Three months after the operation, resuscitation and transplantation of one blastocyst did not result in pregnancy through the natural cycle (NC), followed by resuscitation and transfer of two embryos on day 3 through hormone replacement therapy (HRT), which resulted in a successful pregnancy and live healthy male birth. No recurrence was reported at 19 months after re-FSS. This case highlights the key points of comprehensive multidisciplinary management from the discovery of BOT recurrence, multidisciplinary team (MDT) consultation, ovarian stimulation (OS), egg retrieval, EC, re-FSS, frozen embryo transfer (FET), to delivery. Re-FSS is safe and effective for patients with recurrent serous BOT and strong fertility requirements, and EC before re-FSS is feasible.

Management of cervical cancer in pregnancy in a low resource setting: a rare case report

Cervical cancer in pregnancy is a rare event. Diagnosis and management of cervical cancer in pregnancy is complicated and challenging in a low resource setting. Herein, we present a case of cervical cancer (FIGO stage IB3) diagnosed at 28 This is the first case report on cervical cancer in pregnancy from Bhutan. It highlights the diagnostic and management challenges in a low resource country.

Severe intrahepatic cholestasis of pregnancy due to a Sertoli-Leydig cell tumour in a woman with polycystic ovary syndrome: a case report

Abstract Background Intrahepatic cholestasis of pregnancy (ICP) is a common gestational complication characterized by pruritus and elevated bile acids, usually occurring in the third trimester when the serum estrogen and progesterone levels are highest. Hyperandrogenism during pregnancy is a pathological state that is mostly induced by polycystic ovary syndrome (PCOS) but rarely by concomitant androgen-secreting ovarian tumours. To date, no correlation has been drawn between ICP and hyperandrogenism. Case presentation Here, we present a rare case of early-onset severe ICP in a PCOS patient conceived via in vitro fertilization-embryo transfer, with worsening hirsutism and acne due to high levels of testosterone and dehydroepiandrosterone sulphate, both of which were produced by a fast-growing ovarian Sertoli–Leydig cell tumour. Her serum estradiol was also very high, which was speculated to be converted from the circulating androgens by the placenta. She had preterm premature rupture of membranes and delivered at 30 weeks, followed by a rapid remission of ICP as her serum estradiol dropped. However, the excessive androgens did not retreat until the large ovarian tumour was surgically removed. Conclusion This unusual case highlights the concurrence of original hyperandrogenism and subsequent hyperestrogenism during pregnancy and the resultant confounding manifestations. Obstetricians should be aware of the potential association between androgen excess and ICP via placental aromatization.

Fast-growing immature ovarian teratoma during pregnancy: a case report and a review of the literature

Abstract Background Immature ovarian teratoma is one of the three common malignant ovarian germ cell tumors. However, immature ovarian teratoma in pregnancy is very rare. Due to the rare occurrence, there is little evidence regarding its diagnosis, optimal management, and prognosis. Hence, we present a case of immature teratoma diagnosed during pregnancy, and analyze its clinicopathological features, management and prognosis. Case presentation A 28-year-old woman underwent a sonographic examination revealed no abnormality in the bilateral adnexal area before 29 weeks gestational age (WGA). At 29 WGA, ultrasound demonstrated a 9.7 × 8.5 × 6.4 cm complex structure in the left adnexal area. At 30 WGA, repeated ultrasound revealed rapid growth of tumor mass, measuring 25.0 × 15.0 × 13.7 cm. An elective cesarean section combined with exploratory laparotomy was performed at 33 WGA. Intraoperative frozen pathological examination suggested left ovarian immature teratoma. Then, she underwent a complete staging surgery. Subsequently, the patient received 4 cycles of bleomycin-etoposide-cisplatin (BEP) chemotherapy. After 18 months of follow-up, there is no sign of tumor recurrence till now. Conclusions This case report suggests that the benefits and risks of timely treatment for patients and fetuses should be fully assessed by a multidisciplinary team. The early diagnosis, the timing of surgery and chemotherapy, the choice of chemotherapy for BEP will determine the prognosis. Surgery and combination chemotherapy with BEP play an important role in the treatment of immature teratomas in pregnancy, and could gain successful and satisfactory outcomes for mother and fetus.

Primitive neuroectodermal tumor of the cervix diagnosed during pregnancy: a rare case report with discussion

Abstract Background Primitive neuroectodermal tumor (PNET) is a relatively rare malignant small round cell tumor, and the occurrence of cervical PNET during pregnancy is extremely rare. Case presentation A case of pregnancy complicated by PNET at our hospital was reported. A 19-year-old pregnant woman presented to the hospital due to multiple instances of vaginal bleeding during the first and second trimesters. She was initially considered for threatened abortion but was ultimately diagnosed with cervical PNET. No standard treatment plan has been developed for pregnant women with this tumor. After completing the necessary examinations, doctors cooperated with the patient and her family to develop a surgical treatment plan. The patient recovered well after surgery, but she refused radiotherapy and chemotherapy. After nearly 3 years of follow-up visits, the patient is alive with no signs of recurrence. Conclusions PNET during pregnancy is a rare but complex condition. It is necessary to devise an individualized treatment plan according to gestational age. Timely surgical treatment can significantly prolong the survival time of patients but may also lead to fetal loss and the inability to carry a pregnancy.

Vulvar cancer during pregnancy and/or breastfeeding: a report of five cases from a single center study at the University Hospital of Düsseldorf

Abstract Background The incidence of vulvar cancer (VC) in pregnancy is unknown due to its rarity; between 1955 and 2014 only 36 case reports were reported worldwide. Underreporting may also be a contributing factor to the unknown incidence of VC in pregnancy. The aim of this study was to analyze the diagnosis, treatment and outcome of vulvar cancer cases diagnosed during pregnancy and/or breastfeeding. Case presentation Patient 1 was diagnosed at 18 weeks’ gestation (WG) with Grade 2 VC (pT1a, pN0, 0/4 sentinel lymph nodes biopsy (SLNB) involved) and was treated by having the tumor resected (R0). She is currently recurrence-free at 4 years post-diagnosis. Patient 2 was diagnosed at 7 WG with Grade 2 VC (pT1b, pN1a, 1/17 SLNB, R0) and was treated during the first trimester and during the second trimester with SLNB. She is currently recurrence-free at 5 years post-diagnosis. Patient 3 was diagnosed at 30 WG with Grade 2 VC (pT1b, pN0, 0/5 SLNB, R0). She subsequently experienced a number of local recurrences postpartum that were managed by resection and is currently recurrence-free at 3 years post-diagnosis. Patient 4 was diagnosed a VL later, at 14 months during breastfeeding, that was diagnosed as Grade 3 VC (pT1b, pN1a, 1/14 SLNB, R0). The patient is currently recurrence-free at 9 years post-diagnosis. Patient 5 was not diagnosed during pregnancy, but was diagnosed with G3 VC (pT2, pN2c, 2/17 SLNB, R0) 8 months postpartum. The patient due to the extent of tumor involvement and lymph node metastasis, underwent chemoradiation therapy post-surgery. Despite adjuvant therapy, the patient progressed and developed bone metastases. Analysis of the tumour tissue revealed increased expression of PD-L1 (programmed cell death protein 1) indicating that the patient may have benefited from treatment with nivolumab to block the PD-L1 interaction; unfortunately the patient passed away at 24 months post-diagnosis before immunotherapy treatment could commence. Conclusion Surgical resection and simultaneous SLNB in VC cases are considered safe during pregnancy, with comparable outcomes to non-pregnant women. Prompt diagnostic workup and treatment should never be delayed during pregnancy as delayed diagnosis could lead to tumour progression with fatal consequences.

PET/MR imaging for the evaluation of cervical cancer during pregnancy

Abstract Background Malignancy during pregnancy is increasing, and the most common type of malignancy is uterine cervical cancer. When planning the treatment of cervical cancer, it is important to look for signs of metastasis before surgery, especially metastasis to the lymph nodes. In this report, we assessed the diagnostic value of positron emission tomography/magnetic resonance imaging (PET/MRI) for evaluating cervical cancer propagation before surgery, with a focus on pregnant women. Case presentation 18 F Fluorodeoxyglucose ( FDG)-PET/MRI was performed in seven pregnant cervical cancer patients (28–34 years old) at 9–18 gestational weeks. In case #5, a second PET/MRI was performed at 24 gestational weeks. Of seven FDG-PET/MRI examination series in six cases (cases #1–6), FDG-PET/MR imaging could detect cervical tumors with abnormal FDG accumulation; these tumors were confirmed with a standardized uptake value max (SUV max) titer of 4.5–16. A second PET/MRI examination in case #5 revealed the same SUV max titer as the first examination. In these six imaging series (cases #1–5), there were no signs of cancer metastasis to the parametrium and lymph nodes. However, in case #6, abnormal FDG accumulation in the left parametrial lymph nodes was also detectable. Pathological examination showed lymph node metastasis in case #6. In case #7, PET/MRI could not detect any abnormal FDG accumulation in the cervix and other sites. Cone biopsy demonstrated only micro-invasive squamous cell carcinoma. After treatment for cervical cancer, all seven patients have had no recurrence of disease within the follow-up period (2.8–5.6 years), and their children have developed appropriately. Conclusion PET/MRI is an effective imaging tool to evaluate cervical cancer progression in pregnancy.