Background: Metastatic vulvar choriocarcinoma, a rare ectopic gestational trophoblastic neoplasia (GTN), often presents a diagnostic challenge due to its mimicry of other conditions, particularly in resource-limited settings. Its primary symptom is abnormal vaginal bleeding without a clear cause. Consequently, diagnosing and managing it poses difficulties for many low-resource health facilities, as evidenced by the current case. Case Presentation: We present the case of a 25-year-old, P2+2+2L2, who had a large painless, bleeding vulva mass for nearly 5 months. This followed a spontaneous abortion the month prior. The mass gradually increased in size and was accompanied by fever, pus discharge, and weight loss. Despite being treated at multiple health facilities for a vulvar abscess, there was no improvement. A diagnosis was finally made at a tertiary facility where elevated quantitative serum beta-human chorionic gonadotropin (hCG) (β-hCG) was noted. Due to uncontrollable vulva bleeding, she was referred to another tertiary facility for emergency radiotherapy. Following stabilization, chemotherapy was administered using the EMA-CO protocol. Conclusion: The report highlights the difficulty in diagnosing vulvar choriocarcinoma, underscoring the importance of a high index of suspicion. Clinical tests such as serum (β-hCG) and imaging studies are crucial for diagnosis. In resource-limited settings, a simple strip-based urine pregnancy test with serial dilutions can be sufficient for diagnosing and managing vulvar choriocarcinoma.

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.

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.

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.

UNASSIGNED: In young women presenting with atypical features of intracerebral hemorrhage, metastatic choriocarcinoma should be considered as a differential diagnosis. In resource-poor settings, a high index of suspicion and serum β-hCG are crucial for diagnosis.
UNASSIGNED: Intracerebral hemorrhage in the young is rarely caused by metastatic choriocarcinoma. Diagnosis of this condition may be particularly challenging in resource-poor settings where access to diagnostic technologies may be limited. We present a case series of three young females diagnosed with metastatic choriocarcinoma after initially presenting with intracerebral hemorrhage, each demonstrating unique clinical manifestations. We aim to highlight the diagnostic considerations in the management of this infrequently encountered cause of intracerebral hemorrhage, especially in resource-constrained settings. Case 1 involved a 21-year-old woman who was initially diagnosed with intracerebral hemorrhage likely of tumoral origin from an unknown primary source. Further evaluation revealed extremely high levels of β-hCG and features suggestive of an intrauterine malignancy, which led to a diagnosis of metastatic choriocarcinoma. This further became complicated by pulmonary embolism. Unfortunately, she succumbed to respiratory failure during treatment. Case 2 is a young woman who presented to the emergency unit and was managed as a case of lobar intracerebral hemorrhage. Further checks revealed a previous history of hysterectomy done on account of placental site trophoblastic tumor, which promoted an evaluation for choriocarcinoma. Case 3 involved a 20-year-old patient who initially presented with headache and vomiting. An enhanced magnetic resonance imaging showed a large subacute right temporal occipital subependymal hemorrhage with mass effect. After probing further, we discovered that she underwent exploratory laparotomy for suspected ruptured ectopic gestation, which later turned out to be a gestational trophoblastic neoplasia. After further evaluation a diagnosis of choriocarcinoma with brain metastasis. Our case series emphasizes the importance of having a high index suspicion in young females who present with atypical features of ICH. The varied clinical scenarios highlight the challenges in diagnosing young females. It also underscores the critical role of serum β-hCG, especially in resource-limited settings where biopsies are not readily available. Building a repository of these diverse manifestations is essential for increasing the index of suspicion and ultimately improving patient outcomes.

BACKGROUND: Chemotherapy is crucial in treating gestational trophoblastic neoplasia (GTN), but its impact on gonadotoxicity is unclear.
METHODS: This case-control study included 57 GTN patients and 19 age-matched patients with molar pregnancies (MP) in 2012-2018. Multiples of the median (MoM) of the serum AMH levels were compared between the two groups, and between patients using single-agent and combination chemotherapy, at baseline, 6, 12, and 24 months after treatment. Their pregnancy outcomes were also compared.
RESULTS: There was no significant difference in the MoM of serum AMH between GTN and MP groups at all time points. Single-agent chemotherapy did not adversely affect the MoM. However, those receiving combination chemotherapy had lower MoM than those receiving single-agent chemotherapy at all time points. The trend of decline from the baseline was marginally significant in patients with combination chemotherapy, but the drop was only significant at 12 months (Z = -2.69, p = 0.007) but not at 24 months (Z = -1.90; p = 0.058). Multivariable analysis revealed that combination chemotherapy did not affect the MoM. There was no significant difference in the 4-year pregnancy rate and the livebirth rate between the single-agent and combination groups who attempting pregnancy, but it took 1 year longer to achieve the first pregnancy in the combination group compared to the single-agent group (2.88 vs. 1.88 years).
CONCLUSIONS: This study showed combination chemotherapy led to a decreasing trend of MoM of serum AMH especially at 12 months after treatment, but the drop became static at 24 months. Although pregnancy is achievable, thorough counseling is still needed in this group especially those wish to achieve pregnancy 1-2 years after treatment or with other risk factors.

•Methotrexate (MTX) hypersensitivity is rare and has not been widely reported in the setting of treatment of GTN.•Work up of hypersensitivity reactions may include consultation to an allergist, serum tryptase level, and possible skin testing.•In low-risk GTN, dactinomycin should be utilized after a hypersensitivity reaction to MTX.

UNASSIGNED: Single-agent chemotherapy typically has curative outcomes in patients with low-risk gestational trophoblastic neoplasia (GTN). Although surgical intervention is a potential alternative, its efficacy in these patients remains unclear. This report describes a case in which surgical excision of a uterine polypoid lesion resolved chemotherapy-resistant low-risk GTN.
UNASSIGNED: A 43-year-old patient received pulse actinomycin D treatment for post-molar low-risk GTN without extrauterine metastasis. However, the patient showed resistance to the chemotherapy regimen. There was no initial evidence of protrusion of GTN into the uterine cavity; however, a polypoid lesion grew into the uterine cavity during therapy. This growth was successfully excised via a transvaginal approach using forceps with minimal blood loss. There was a postoperative decrease in human chorionic gonadotropin levels, which ultimately reached the predetermined threshold without the need for changing the therapeutic protocol.
UNASSIGNED: Surgical resection should be considered a viable therapeutic strategy for uterine polypoid growth in chemotherapy-resistant low-risk GTN.