Molar pregnancy with a coexisting living fetus: a case series.

BACKGROUND: Coexistence of molar pregnancy with living fetus represents a challenge in diagnosis and treatment. The objective of this study to present the outcome of molar pregnancy with a coexisting living fetus who were managed in our University Hospital in the last 5 years. METHODS: We performed a retrospective analysis of patients who presented with molar pregnancy with a coexisting living fetus to our Gestational Trophoblastic Clinic, Mansoura University, Egypt from September, 2015 to August, 2020. Clinical characteristics of the patients, maternal complications as well as fetal outcome were recorded. The patients and their living babies were also followed up at least 6 months after delivery. RESULTS: Twelve pregnancies were analyzed. The mean maternal age was 26.0 (SD 4.1) years and the median parity was 1.0 (range 0-3). Duration of the pregnancies ranged from 14 to 36 weeks. The median serum hCG was 165,210.0 U/L (range 7662-1,200,000). Three fetuses survived outside the uterus (25%), one of them died after 5 months because of congenital malformations. Histologic diagnosis was available for 10 of 12 cases and revealed complete mole associated with a normal placenta in 6 cases (60%) and partial mole in 4 cases (40%). Maternal complications occurred in 6 cases (50%) with the most common was severe vaginal bleeding in 4 cases (33.3%). There was no significant association between B-hCG levels and maternal complications (P = 0.3). CONCLUSION: Maternal and fetal outcomes of molar pregnancy with a living fetus are poor. Counseling the patients for termination of pregnancy may be required. TRIAL REGISTRATION: The study was approved by Institutional Research Board (IRB), Faculty of Medicine, Mansoura University (number: R.21.10.1492).

Ovarian Hyperstimulation Syndrome Complicating Spontaneous Molar Pregnancy: A Case Report and Review of the Literature.

BACKGROUND: Ovarian hyperstimulation syndrome (OHSS) is traditionally associated with fertility treatments and results in elevated human chorionic gonadotropin (ßhCG) levels and fluid shifts to extravascular compartments. Rarely, spontaneous pregnancies with significant ßhCG elevations, such as molar pregnancies, can give rise to OHSS. CASE: A 24-year-old woman was diagnosed as having a molar pregnancy at approximately 12 weeks gestation following spontaneous conception. Her initial ßhCG was over 1 million IU/L. There was no evidence of metastatic disease. She underwent an uncomplicated dilation and curettage. Three days later, she presented with chest pain, shortness of breath, and abdominal discomfort. Massively enlarged ovaries were identified with bilateral pleural effusions requiring repeated thoracentesis. CONCLUSION: This case demonstrates rare sequelae of molar pregnancy. Treatment is mainly supportive, and close observation is required to manage complications. In patients with extremely elevated ßhCG levels, clinicians must remain vigilant for signs suggesting OHSS, even following evacuation of the uterus.

Histological Examination of Tissue Obtained in Early Pregnancy Loss.

Background It is a routine practice to send histological sample after surgical evacuation of early pregnancy loss. Objective This study was carried out to see the justification of regular histological study by carrying out the histological study of early pregnancy loss and to find the prevalence of gestational trophoblastic disease in early pregnancy loss. Method It was a descriptive prospective study, conducted in Nepal medical college teaching hospital from February to October 2020 in Obstetrics and Gynaecology department. Clinical data such as age, parity, gestational age and diagnosis were collected of 130 patient of early pregnancy loss. Then histological study were sent after surgical evacuation. Result Among the age group, 21-30 age group was maximum. (64.61%), more than half of the patient was primigravida (53.07%) and most of the cases were between 6 to 9 weeks of gestation. Incomplete abortions were maximum (43.07%), missed abortions 38.46%, blighted abortions 16.15%, enevitable abortions 1.53% and septic abortion was 0.76%. Among histological finding, 72.30% were product of conception, 15.38% of the cases had no product of conception, decidual tissue only in 6.92%, partial mole in one case (0.76%), complete mole in one case (0.76%) and hydrophic changes in one case (0.76%). The total cases of Gestational trophoblastic diseases (GTD) were 3(2.30%). Conclusion In our study we found 2.3% of cases of GTD, which was quite high in compare to Western word. So it is a good practice to do histological study of all cases of EPL in our country to detect GTD, determining cause for recurrent pregnancy loss and detecting unexpected fetal pathology.

Evaluation of a routine second curettage for hydatidiform mole: a cohort study.

OBJECTIVE: The aim of this study was to evaluate routine second curettage for hydatidiform mole (HM) by comparing the characteristics and outcomes of developing gestational trophoblastic neoplasia (GTN). STUDY DESIGN: This was a cohort study including 173 patients diagnosed with HM between January 2002 and August 2019 who were followed up at Nagoya University Hospital, Japan. After an evacuation, 105 and 68 patients were managed with the routine method (routine group) and elective method (elective group) for a second curettage, respectively. The routine second curettage was performed around 7 days after the first evacuation. Patients in the elective group underwent a second curettage if there was ultrasonographic evidence of molar remnants in the uterine cavity. Socio-clinical factors were retrospectively compared between the routine and elective groups, and between patients showing regression and those who developed GTN. RESULTS: The incidence of GTN was 15.2% in the routine group and 20.6% in the elective group, and the difference was not significant (P = 0.364). The median GTN risk score was significantly higher in the routine group than in the elective group (P = 0.033). Presence of a complete HM, gestational age, and a pre-treatment human chorionic gonadotropin level of ≥ 200,000 mIU/mL were independent risk factors for GTN in molar patients. CONCLUSION: The incidence of GTN was unchanged but the risk score of GTN was higher in the routine group than in the elective group. Routine second curettage may not be necessary, but further study will be needed to confirm this.

Prenatal Diagnosis of Twin Pregnancies with Complete Hydatidiform Mole and Coexistent Normal Fetus: A Series of 13 Cases.

AIM: The study aimed to describe prenatal diagnosis and the outcome of complete hydatidiform mole and coexistent normal fetus (CHMCF). METHODS: This was a retrospective case series of 13 patients with CHMCF. Prenatal diagnosis, outcome and development of gestational trophoblastic neoplasia (GTN) were reviewed. RESULTS: Ultrasound diagnosis was carried out in 12 of 13 cases at 17 ± 2.7 weeks of gestation (mean ± SD). Six patients showed abnormalities suggestive of subchorionic hematoma on first trimester ultrasonography (US). Prenatal invasive procedures were performed in 8 of 13 cases (62%). Two women decided to terminate their pregnancies. Four ended in late miscarriages (36%, 4 of 11) between 13 and 21 weeks, and early neonatal death occurred in 1 case (9%, 1 of 11); 5 women delivered a live baby with a mean gestational age of 31 weeks (range 26-37 weeks) with an overall neonatal survival of 45% (5 of 11). GTN occurred in 31% of cases (4 of 13). CONCLUSIONS: The first trimester US features of CHMCF are not well-documented. Our series showed that abnormalities of CHMCF could be misdiagnosed as subchorionic hematoma in the early first trimester. When CHMCF is confirmed by expert US, prenatal invasive procedures should be carefully evaluated depending on the associated US findings and exhaustive counseling should be performed.

Unusual twin pregnancy: complete hydatidiform mole with coexistent normal fetus.

The authors present a case of complete hydatidiform mole and coexisting fetus (CHMCF) in which mole gestation caused a placenta previa; with a posterior preterm premature rupture of membranes (PPROM) and ending in the 28h week of gestation due to acute chorioamnionitis, obtaining a live preterm newborn.

[Twin pregnancy with complete hydatidiform mole]. / Polnyi puzyrnyi zanos pri beremennosti dvoinei.

The paper describes a case of twin pregnancy with complete hydatidiform mole (CHM). According to the data available in the literature, the concurrence of CHM with a normal placenta and a viable fetus occurs in 1 per 20,000-100,000 pregnancies, requires a differential diagnosis with partial hydatidiform mole and placental mesenchymal dysplasia, and is characterized by a high rate of complications. In this concurrence, the frequency of persistent trophoblastic disease is as high as 50%. In this case, the pregnancy ended in a spontaneous abortion at 16-17 weeks of pregnancy. A morphological examination determined the fetus without congenital malformations with normal placental weight and structure and the adjacent intact placental tissue with the macro- and microscopic signs of CHM. The diagnosis was confirmed by the lack of р57 expression in the villous trophoblast and stroma in the tissue of the hydatidiform mole. The patient was diagnosed with persistent trophoblastic disease at 2 months after the abortion.

Ultrasound diagnosis and management of acquired uterine enhanced myometrial vascularity/arteriovenous malformations.

BACKGROUND: Arteriovenous malformation is a short circuit between an organ's arterial and venous circulation. Arteriovenous malformations are classified as congenital and acquired. In the uterus, they may appear after curettage, cesarean delivery, and myomectomy among others. Their clinical feature is usually vaginal bleeding, which may be severe, if curettage is performed in unrecognized cases. Sonographically on 2-dimensional grayscale ultrasound scanning, the pathologic evidence appears as irregular, anechoic, tortuous, tubular structures that show evidence of increased vascularity when color Doppler is applied. Most of the time they resolve spontaneously; however, if left untreated, they may require involved treatments such as uterine artery embolization or hysterectomy. In the past, uterine artery angiography was the gold standard for the diagnosis; however, ultrasound scanning has diagnosed successfully and helped in the clinical management. Recently, arteriovenous malformations have been referred to as enhanced myometrial vascularities. OBJECTIVES: The purpose of this study was to evaluate the role of transvaginal ultrasound scanning in the diagnosis and treatment of acquired enhanced myometrial vascularity/arteriovenous malformations to outline the natural history of conservatively followed vs treated lesions. METHODS: This was a retrospective study to assess the presentation, treatment, and clinical pictures of patients with uterine Enhanced myometrial vascularity/arteriovenous malformations that were diagnosed with transvaginal ultrasound scanning. We reviewed both (1) ultrasound data (images, measured dimensions, and Doppler blood flow that were defined by its peak systolic velocity and (2) clinical data (age, reproductive status, clinical presentation, inciting event or procedure, surgical history, clinical course, time intervals that included detection to resolution or detection to treatment, and treatment rendered). The diagnostic criteria were "subjective" with a rich vascular network in the myometrium with the use of color Doppler images and "objective" with a high peak systolic velocity of ≥20 cm/sec in the vascular web. Statistical analysis was performed and coded with statistical software where necessary. RESULTS: Twenty-seven patients met the diagnostic criteria of uterine enhanced myometrial vascularity/arteriovenous malformation. Mean age was 31.8 years (range, 18-42 years). Clinical diagnoses of the patients included 10 incomplete abortions, 6 missed abortions, 5 spontaneous complete abortions, 5 cesarean scar pregnancies, and 1 molar pregnancy. Eighty-nine percent of patients had bleeding (n = 24/27), although 1 patient was febrile, and 2 patients were asymptomatic. Recent surgical procedures were performed in 55.5% patients (15/27) that included curettage (n = 10), cesarean deliveries (n = 5), or both (n = 1); 4 patients had a remote history of uterine surgery that included myomectomy. Treatment was varied and included expectant treatment alone in 48% of the patients with serial ultrasound scans and serum human chorionic gonadotropin until resolution (n = 13/27 patients), uterine artery embolization (29.6%; 8/27 patients), methotrexate administration (22.2%; 6/27 patients), hysterectomy (7.4%; 2/27 patients), and curettage (3.7%; 1/27 patients). Three patients required a blood transfusion. Of the 9 patients whose condition required embolization, the conditions of 7 patients resolved after the procedure although 1 patient's condition required operative hysteroscopy and 1 patient's condition required hysterectomy for intractable bleeding. Average peak systolic velocity after embolization in the 9 patients was 85.2 cm/sec (range, 35-170 cm/sec); the average peak systolic velocity of the 16 patients with spontaneous resolution was 58.5 cm/sec (range, 23-90 cm/sec). CONCLUSIONS: Acquired enhanced myometrial vascularity/arteriovenous malformations occurred after unsuccessful pregnancies or treatment procedures that included uterine curettage, cesarean delivery, or cesarean scar pregnancy. Triage of patients for expectant treatment vs intervention with uterine artery embolization based on their clinical status, which was supplemented by objective measurements of blood velocity measurement in the arteriovenous malformation, appears to be a good predictor of outcome. Ultrasound evaluation of patients with early pregnancy failure and persistent bleeding should be considered for evaluation of a possible enhanced myometrial vascularity/arteriovenous malformation.

An unusual cause of cardiothyreosis.

Severe hyperthyroidism can cause cardiac complications, such as severe rhythm disturbances, heart failure and angina. Gestational trophoblastic disease (GTD) is a rare complication of pregnancy, ranging from benign hydatidiform mole to malignant form. Clinical hyperthyroidism may occur in GTD, as human chorionic gonadotropin (hCG) secreted by molar tissue is structurally similar to thyroid-stimulating hormone. Cardiothyreosis in this context is exceptional. We report the case of a nulligravida 42-year-old woman without thyroid or cardiac history who presented to the emergency department for dyspnoea. Examinations revealed an acute pulmonary oedema and sinus tachycardia. Serum hCG concentration was abnormally high (762 878 UI/l, N < 5). CT scan showed a voluminous uterine mass and eliminated pulmonary embolism. Cardiac output was increased in echocardiography. Complementary blood tests showed a peripheral hyperthyroidism. GTD was evoked in the context of uterine mass and high hCG concentration, which was responsible for inducing clinical hyperthyroidism and cardiothyreosis. A total hysterectomy was performed and histopathological examinations concluded to a non-invasive complete hydatidiform mole (begnin form). hCG fell to normal within 12 weeks, cardiac and thyroid functions normalized after mole evacuation.

Diginyc partial hydatidiform mole with increased fetal nuchal translucency and ovarian hyperstimulation syndrome.

PURPOSE OF INVESTIGATION: Hydatidiform mole (HM) is an abnormal pregnancy characterized by proliferation of cytotrophoblast and syncytiotrophoblast and vesicular swelling of placental villi. The fetus or embryo can be absent or abnormal. HMs can be complete or partial. CASE REPORT: A case of diginyc partial HM at 12 weeks of gestational age was referred to the present center of prenatal diagnosis. The patient showed ovarian hyperstimulation syndrome. At ultrasonography, increased fetal nuchal translucency (NT) with fetal anomaly was evident, without sonographic signs of placental mole. Pregnancy was terminated with legal abortion. RESULTS: Partial HM (PHM) was suspected by ultrasonographic fetal markers with ovarian hyperstimulation syndrome, but the diagnosis was performed only with fluorescent in situ hybridization. In particular fetal NT appeared increased also in diginyc mole. CONCLUSION: In order to improve the detection rate of PHM, routine histological examinations may be associated to fluorescent in situ hybridization in all cases of fetal anomalies.

Live births in women with recurrent hydatidiform mole and two NLRP7 mutations.

Hydatidiform mole (HM) is an aberrant human pregnancy with abnormal embryonic development and excessive proliferation of the trophoblast. Recessive mutations in NLRP7 are responsible for recurrent HM (RHM). Women with recessive NLRP7 mutations fail to have normal pregnancies from spontaneous conceptions with the exception of three out of 131 reported patients. Because there is no treatment for RHM and maternal-effect genes are needed in the oocytes to sustain normal embryonic development until the activation of the embryonic genome, one patient with recessive NLRP7 mutations tried ovum donation and achieved a successful pregnancy. This study reports three additional live births from donated ova to two patients with recessive NLRP7 mutations. The occurrence of two live births from spontaneous conceptions to two other patients is also reported. The reproductive outcomes and mutations of all reported patients were reviewed and it was found that live births are associated with some missense mutations expected to have mild functional consequences on the protein. The data support a previous observation that ovum donation appears the best management option for these patients to achieve normal pregnancies and provide an explanation for the rare occurrence of live births from natural spontaneous conceptions in patients with two NLRP7 mutations.

Thyroid storm-induced multi-organ failure in the setting of gestational trophoblastic disease.

BACKGROUND: Thyroid storm is a potentially life-threatening complication of gestational trophoblastic disease (GTD), with varying clinical severity. It should be considered in patients with GTD, abnormal vital signs, and clinical signs of hyperthyroidism. CASE REPORT: A 45-year-old non-English-speaking patient presented to a New York City hospital in November 2011 with an aborting molar pregnancy and severe hemorrhage. Initial presentation was concerning for GTD. Laboratory values were obtained that confirmed the diagnosis of GTD, which was also by thyroid storm and congestive heart failure. This was evidenced by laboratory values of free thyroxine of 4.9 and beta human chorionic gonadotropin of 1,488,021 IU/mL. Dilation and curettage with 16-mm suction catheter was performed until all products of conception were removed and bleeding was controlled. The patient was admitted to the surgical intensive care unit and proceeded to have multi-organ failure, and remained intubated and unresponsive to verbal/visual and tactile stimuli. On postoperative day 13 the patient suddenly became alert and self-extubated, began to communicate verbally, and resolution of her multi-organ failure became evident. The patient was discharged with Gynecologic Oncology follow-up. Why should an emergency care physician be aware of this? This case represents the dangers associated with poor prenatal care and late diagnosis of molar pregnancy. It also represents the need for immediate recognition of the condition and initiation of appropriate medical care. Although this patient's clinical outcome was good, the event could have been prevented had she received reliable medical care.

Clostridium perfringens sepsis following a molar pregnancy.

Clostridium perfringens sepsis is rare since the legalization of abortion in 1973. This is a 49 year old female who developed clostridial sepsis after suction dilation and curettage for a molar pregnancy. A hysterectomy was performed after prompt recognition, and the patient survived.

Women with a partial mole during their first pregnancy and diagnosed earlier in gestation are at increased risk of developing gestational trophoblastic neoplasia.

OBJECTIVE: The aim of this study is to identify factors associated with gestational trophoblastic neoplasia (GTN) after partial molar pregnancy. METHODS: We retrospectively evaluated clinical data from 111 patients with a partial molar pregnancy between 1995 and 2010. RESULTS: A total of 111 patients with a partial molar pregnancy were available for analysis. There was no significant difference between patients who did and did not develop GTN with respect to patient age, parity, history of prior molar pregnancy, presenting signs/symptoms, uterine size greater than gestational age, clinical diagnosis, preevacuation sonogram findings, or the preevacuation human chorionic gonadotropin value. Patients who developed GTN had fewer prior pregnancies (median, 2 vs 3; P = 0.02) and were more likely to have had a partial molar pregnancy as their first gestational event (37.1% vs 17.1%; P = 0.03). Among the 35 patients who developed GTN, the median time to diagnosis of GTN was 47 days (range, 25-119 days), and the median human chorionic gonadotropin value at the time of GTN diagnosis was 475 mIU/mL (range, 20-52,630 mIU/mL). All women (100%) who developed GTN had stage I disease, and all patients (100%) had low-risk GTN. All 35 women (100%) were able to achieve remission, and most (85.7%) of these patients received methotrexate as first-line chemotherapy. CONCLUSIONS: Women with a partial molar pregnancy as their first gestational event and diagnosed earlier in gestation are more likely to develop postmolar GTN.

Hydatidiform mole: age-related clinical presentation and high rate of severe complications in older women.

OBJECTIVE: The purpose of this study was to demonstrate differences in clinical presentation of hydatidiform mole between women ≥40 years and younger women. DESIGN: Retrospective study. SETTING: A tertiary referral unit in northern Italy. POPULATION: Three hundred and sixty-five women with hydatidiform mole were divided into group A (<40 years, 318 cases) and group B (≥40 years, 47 cases). METHODS: Clinical presentation between groups A and B was analyzed, also considering partial hydatidiform mole and complete hydatidiform mole. MAIN OUTCOME MEASURES: Differences in clinical presentation according to woman's age. RESULTS: In group B the diagnosis of hydatidiform mole at ≥12 gestational weeks was more frequent (p < 0.001) and the detection of ultrasound features was higher (p < 0.05) than in group A. Vaginal bleeding (p < 0.05), increased uterine volume (p < 0.0001) and hyperemesis (p < 0.05) occurred more frequently in group B. In the women with complete hydatidiform mole, group B women presented with vaginal bleeding (p < 0.001), increased uterine volume (p < 0.05) and hyperemesis (p < 0.05) more frequently than group A women. Complete hydatidiform mole was more commonly diagnosed after 12 weeks of gestation in group B (p < 0.0001). In women ≥50 years, an increased rate of disease-related complications was detected. CONCLUSIONS: The clinical features of hydatidiform mole in women ≥40 years are different from those seen in younger women. Failures in the early detection of hydatidiform mole in older women may expose them to a higher rate of severe complications.