OBJECTIVE: To elucidate the clinicopathological characteristics and oncological outcomes of a special group of patients with gestational trophoblastic neoplasia (GTN) initially presenting with isolated lung lesions, elevated human chorionic gonadotropin (hCG) levels, and unobserved pelvic lesions.
METHODS: Overall, 2358 patients with GTN treated at our hospital between 2000 and 2023 were retrospectively reviewed, and 40 patients were evaluated. The demographic characteristics, clinicopathological features, treatment data, and follow-up information of each patient were collected. The primary outcome was progression free survival. Kaplan-Meier analysis and univariate and multivariate Cox proportional hazard analyses were used to identify the risk factors.
RESULTS: Among the 40 patients, 95.0 % had solitary lung lesions, with a median size of 1.9 cm. Moreover, 72.5 % of patients were pathologically confirmed as epithelioid trophoblastic tumors (ETT). During a median follow-up period of 53.5 months (range, 2-143), 11 patients experienced recurrence, including all patients who received chemotherapy alone as the initial treatment, and no death was observed. Relapse treatment involved lung segmentectomy and lobectomy combined with chemotherapy and immunotherapy. Univariate and multivariate Cox analyses identified comparing with surgery±chemotherapy, chemotherapy alone as the initial treatment (hazard ratio [HR] =7.738, 95 % confidence interval [CI] 1.698-35.269, P = 0.008) as independent risk factor for recurrence.
CONCLUSIONS: In patients with a history of pregnancy exhibiting isolated pulmonary lesions, elevated hCG levels (mostly <1000 mIU/mL), and unobserved pelvic lesions, ETT should be considered first. Surgical resection of lung lesion is crucial for optimal management. When chemotherapy is considered, multidrug regimen is recommended.

Gestational trophoblastic neoplasia (GTN) with spinal metastasis is rare with few documented cases worldwide. Few studies have explored chemotherapy combined with radiotherapy in the treatment of such cases. However, because of its rarity, there is still no standardized treatment regimen. A 34-year-old Gravida 1 Para 0 (0010) was diagnosed with GTN with metastasis to the lumbosacral spine, resulting in conus medullaris syndrome with lumbar radiculopathy. She presented with a 14-month history of amenorrhea, left lower extremity pain, and urinary and bowel retention. On examination, there was a 10.0 × 7.0 cm lumbosacral mass and atrophy of the left lower extremity. Transvaginal ultrasound showed a cul de sac mass, and diluted β-human chorionic gonadotropin (β-hCG) titer was markedly elevated at more than 1000 000 mIU/mL. Magnetic resonance imaging (MRI) of the lumbosacral spine showed an ill-defined sacral mass measuring 13.3 × 11.5 × 6.3 cm with spinal canal, bone, muscle, and nerve root involvement. She was treated with 10 cycles of EMACO and palliative radiotherapy with 10 sessions of 30 Gy of external beam radiation therapy directed toward the lumbosacral mass. Repeat MRI showed a decrease in size of the mass to 6.6 × 8.2 × 4.1 cm with concurrent decrease in β-hCG to 1.30 mIU/ml, and resolution of leg pain and urinary and bowel symptoms. She was declared to be in remission 3 months after the last cycle of EMACO.

UNASSIGNED: In addressing Gestational Trophoblastic Neoplasia (GTN), it is imperative to acknowledge the evolving landscape of treatment options, especially in light of the challenges posed by traditional methods. While historically, surgical interventions, radiation therapy, and chemotherapeutic agents have been the mainstays, the emergence of resistance and high-risk scenarios necessitates a reevaluation of our therapeutic approaches. Our review highlights the promising advancements in immunotherapy and molecular targeted therapy as viable alternatives for GTN management. The introduction of immune checkpoint inhibitors and kinase inhibitors offers a paradigm shift, particularly for patients resistant to conventional chemotherapy regimens. These novel therapies not only exhibit efficacy but also demonstrate manageable toxicity profiles, particularly in high-risk cases. However, integrating these innovative treatments into established international guidelines presents a formidable task. As we move forward, it is imperative that future research not only prioritizes fertility preservation but also rigorously evaluates long-term toxicity implications. International collaboration becomes pivotal in addressing the nuances of this rare and complex disease. In conclusion, our review underscores the need for a nuanced approach to GTN treatment, one that prioritizes reduced toxicity and improved quality of life. By embracing the advancements in immunotherapy and molecular targeted therapy, we can pave the way for more effective and patient-centered care in the management of GTN.

UNASSIGNED: This study aimed to determine the clinical outcomes of ultra high-risk gestational trophoblastic neoplasia (GTN) patients managed with and without induction chemotherapy in the Division of Trophoblastic Diseases, Department of Obstetrics and Gynecology, Philippine General Hospital.
UNASSIGNED: Clinical and demographic data were collected retrospectively from ultra high-risk GTN patients admitted in the Division of Trophoblastic Diseases, Department of Obstetrics and Gynecology, Philippine General Hospital from January 2015 to December 2021. Rate of remission and early death of those who received induction chemotherapy were compared to those who did not.
UNASSIGNED: A total of 21 patients with ultra high-risk GTN were included in the study, nine of whom underwent induction chemotherapy while 12 had no induction chemotherapy and was given the standard EMACO regimen. There was no significant difference in the rate of early death as well as the rate and time to achieve remission between those who received induction chemotherapy compared to those who were immediately started on EMACO.
CONCLUSIONS: A firm conclusion cannot be drawn from the results considering the small population included in the study. Further studies with larger sample size and prospective study design are recommended.

This is the first reported case of the use of immunotherapy in chemo-resistant Gestational Trophoblastic Neoplasia (GTN) in the country. A 41-year-old, Gravida 4 Para 3 (3013) with a diagnosis of GTN, Stage III: WHO risk score of 13 (Choriocarcinoma) was initially managed with 10 cycles of multiple agent Etoposide, Methotrexate, Actinomycin D-Cyclophosphomide and Vincristine (EMACO) and 19 cycles of Etoposide, Cisplatin-Etoposide Methotrexate and Actinomycin D (EP-EMA). With continuous rise in beta human chorionic gonadotropin (ßhCG) levels, the patient was referred to a Trophoblastic Disease Center where there was note of tumor progression to the brain. She was started on third-line salvage chemotherapy of Paclitaxel and Carboplatin (PC) with concomitant whole brain irradiation completing three cycles after which chemoresistance was again diagnosed with increasing hCG titers and increase in the number and size of the pulmonary masses which were deemed unresectable. Immunotherapy was started with Pembrolizumab showing a good response with marked fall in ßhCG levels. The onset of immune-related adverse events (irAEs) caused a marked delay in subsequent cycles of immunotherapy. With management of the irAEs, two more cycles of Pembrolizumab with fifty percent dose reduction were given with corresponding drop in ßhCG levels. However, the patient subsequently developed gram-negative septicemia with possible hematologic malignancy and finally succumbed to massive pulmonary embolism. The case highlights the importance of prompt diagnosis and referral to a Trophoblastic Disease Center and the use of immunotherapy in chemo-resistant GTN.

The present study reports a rare case of an exaggerated placental site (EPS) in a caesarean scar that was misdiagnosed as gestational trophoblastic neoplasia (GTN) by imaging, resulting in unnecessary surgical treatment. A 38-year-old woman underwent hysteroscopic resection of a cesarean scar pregnancy (CSP). The patient\'s serum β-human chorionic gonadotropin (β-hCG) level was elevated (76,196 mIU/ml) at the 24-day postoperative follow-up visit. On postoperative day 51, the patient experienced vaginal bleeding for three days and β-hCG levels were 2,799 mIU/ml. Ultrasonography and MRI revealed a heterogeneous mass and hypervascularity. The patient was diagnosed with a GTN in a cesarean scar and treated with methotrexate (MTX). β-hCG levels decreased after 3 MTX doses, but the mass did not change in size and was still hypervascular on imaging. Total hysterectomy was performed due to the serious side effects of chemotherapy and the lack of desire to preserve fertility. The histological findings supported the diagnosis of an EPS reaction. The present case is unique because of the rare intrauterine mass and possibility of retained trophoblastic changes causing EPS. EPS differs from GTN both clinically and pathologically and should be considered a possible diagnosis in any woman who has irregular bleeding following CSP resection.

Gestational trophoblastic neoplasia (GTN) is extremely rare, but has a very good prognosis, with a cure rate close to 100%, for low-risk diseases. This article describes the case of a healthy 28-year-old nulliparous patient with GTN resistant to multiple lines of treatment. The era of immunotherapy is revolutionizing oncology, having already proved its worth in the treatment of many cancers. This article will have a specific focus on the emerging role of immunotherapy in the treatment of GTN. Unfortunately, the use of an immune checkpoint inhibitor (ICI) failed in our case, emphasizing on the necessity to clearly define the future role of immune therapy in GTN. Finally, given the rapid progression of the disease after hysterectomy, induction with Paclitaxel- Ifosfamide and then intensification with high-dose Carboplatin and Etoposide with peripheral blood stem cell support was given as a rescue therapy with still curative intent.

BACKGROUND: Intraplacental choriocarcinoma (IC) is an extremely rare subtype of gestational choriocarcinoma. The long-term follow-up and reproductive outcomes of IC patients remain unclear. Here, we report a series of 14 cases and conduct a literature review to assess the fertility and recurrence results of this rare disease.
RESULTS: Fourteen patients with pathologically confirmed IC treated in Peking Union Medical College Hospital between January 2002 and July 2022 were included in this study. Half of them had metastatic IC and were treated by chemotherapy with or without surgery. Only 1 patient had chemoresistant disease, but she achieved complete remission after immunotherapy. The median follow-up time was 45.5 months (range 4-192), and no recurrence occurred. One metastatic IC patient who achieved remission after chemotherapy had a full-term delivery. Among the 5 patients with fertility demands, 3 abandoned their pursuit of pregnancy because of \"fear and worry about choriocarcinoma recurrence\". We reviewed a total of 89 cases of IC in English and Chinese literature from 1963 to 2022, and only 5 cases with subsequent pregnancy were reported, all of them were nonmetastatic IC cases.
CONCLUSIONS: IC is sensitive to chemotherapy and has good long-term remission and a low recurrence rate. Patients with metastatic or nonmetastatic IC can have good pregnancy results after treatment. Doctors should pay more attention to the psychology of these patients.
BACKGROUND: N/A.

Gestational trophoblastic neoplasia (GTN) comprises a group of human neoplastic diseases that derive from fetal trophoblastic tissues. They are proliferative as well as degenerative disorders of placental elements and include complete hydatidiform mole (CHM) or partial hydatidiform mole (PHM) (90%), invasive mole (IM) (5-8%), which could also be metastatic, villous, or villous choriocarcinoma (CC) (1-2%), and placental site trophoblastic tumor (PSTT) (1-2%). We present three cases of GTN, two mimicking tuberculosis radiologically, and all three are associated with pulmonary embolism.

Introduction  Triploid partial molar pregnancies are not viable, and confer maternal risks including preeclampsia, hemorrhage, gestational trophoblastic neoplasia, and trophoblastic embolization. We report a case managed expectantly until 26 weeks\' gestation in a patient requesting continuation of pregnancy. Case   Presentation This G2P1 presented with fetal anomalies indicative of triploid partial molar pregnancy. The pregnancy was complicated by anemia, hyperthyroidism, supraventricular tachycardia, and threatened preterm labor. Her care involved maternal fetal medicine collaborating with internal medicine, palliative care, anesthesia and critical care. Labor was augmented at 26 weeks\' gestation, resulting in vaginal delivery. Postpartum course was notably complicated by acute respiratory distress in the immediate postpartum period, which self-resolved. Postpartum hemorrhage and retained products of conception were additional complications. Conclusion  This unique case highlights the role of multidisciplinary collaboration and shared decision making in challenging circumstances.