Complete hydatidiform mole and a coexistent viable fetus: report of two cases and review of the literature

OBJECTIVE: The aim of this study was to report the clinical features, management, and outcome of two cases of complete hydatidiform mole with a coexisting viable fetus and to review the literature. PATIENTS: In this article, we report on the well-documented follow-up of 2 cases of twin pregnancies with complete hydatidiform mole and a viable fetus, both of which ended with the delivery of a normal infant at 41 and 26 weeks of gestation. It is of interest that both pregnancies were achieved following induction of ovulation with hMG/hCG. Since 1977, the year in which complete and partial moles were characterized as distinct pathologic entities, 15 cases (including our 2) have been reported. RESULTS: Persistent GTT developed in eight patients (53.3%) and four patients (27.7%) developed metastatic disease. Seventy-five percent patients with persistent GTT were treated with single-agent chemotherapy. The median gestational age of the patients with subsequent persistent GTT was 34.5 weeks compared to 38 weeks in the patients without persistent GTT. CONCLUSION: Complete hydatidiform mole and coexistent fetus is a rare occurrence and is associated with an increased risk of persistent gestational trophoblastic tumor. Based on currently available information, it seems that in the presence of a stable pregnancy, normal karyotype, and a normal sonogram it is reasonable to allow the pregnancy to continue.     

Outcome of pregnancies occurring within 1 year of hydatidiform mole.

OBJECTIVE: To determine the outcome of subsequent pregnancies in patients with partial or complete molar pregnancy who conceive before completing the recommended hCG follow-up of at least 6 months. METHODS: Retrospective record review of patients with partial or complete mole who conceived before the standard gonadotropin follow-up of 6 months was completed during 1980-1998. RESULTS: Sixty-seven patients with molar pregnancy who conceived before completion of hCG follow-up were identified. Thirty-five (52.2%) patients had a prior partial mole, and 32 (47.8%) had a prior complete mole. The mean interval from first achieving undetectable hCG level to new pregnancy was 3.1 and 3.4 months in patients with partial and complete mole, respectively. Eleven patients underwent elective termination, and 12 were lost to follow-up. Of the remaining 44 patients, 33 (75.0%) had live births, 10 had spontaneous abortions, and one had an ectopic pregnancy. A viable pregnancy outcome was achieved in 20 (83.3%) of 24 patients with partial mole and 13 (65.0%) of 20 patients with complete mole. None of the patients developed any evidence of postmolar persistent gestational trophoblastic tumor. None of the live births had any detectable fetal anomalies. CONCLUSION: The risk of persistent tumor is low and reproductive outcome is favorable once undetectable hCG levels are achieved. Pregnancies occurring before the completion of recommended hCG follow-up may be allowed to continue under careful surveillance.     

Metastatic complete hydatidiform mole with a surviving coexistent twin. A case report

BACKGROUND: Complete hydatidiform molar pregnancy with a coexistent live fetus is a rare clinical entity with poor fetal survival and an uncertain maternal risk. CASE: A twin gestation consisting of a complete molar pregnancy as one twin and a normal second "twin" was identified in the second trimester. The patient was managed expectantly until 28 weeks' gestation, at which time evidence of metastatic trophoblastic disease was identified. She delivered a healthy infant by cesarean section and received postoperative single-agent chemotherapy. She was disease free six months after treatment. CONCLUSION: Patients with documented complete molar pregnancies and coexistent live fetuses present several complex clinical issues. Though successful in some instances, expectant management should be used with caution in these patients.     

Complete hydatidiform mole and a coexistent fetus following ovulation induction in a patient with Sheehan’s syndrome: a first case report and review of literature

Pregnancy in Sheehan’s syndrome (SS) is extremely rare. We present the first reported case of twin pregnancy with complete hydatiform mole (CHM) and a coexistent fetus (CHCF) in a patient with SS. A 29-year-old Chinese patient with SS became pregnant following one cycle of ovulation induction with human menopausal gonadotropin after secondary infertility. A normal live fetus and a low echogenic mass suspected hydatidiform mole (HM) were detected by ultrasound examinations at gestational week 8. The couple highly desired to continue the pregnancy because it is very hard to get pregnant for the patients with SS. However, the pregnancy was terminated for the size of the HM component increased rapidly at gestational week 15. Histological examinations confirmed CHCF. Genetic studies showed that the CHM genome was derived from paternal diploidy, and the normal fetus was from biparental genomes. Furthermore, a literature review on these topics is included. This case highlighted that even in a patient with SS, twin pregnancy with CHCF can still occur after ovulation induction.     

Complete hydatidiform mole coexisting with a live fetus after gamete intrafallopian transfer. A case report

BACKGROUND: Molar pregnancy with a coexisting live fetus is a rare occurrence. We report the only known case with a surviving coexistent fetus after gamete intrafallopian transfer (GIFT). CASE: After GIFT, a 28-year-old primary infertility patient was diagnosed as having a complete hydatidiform mole coexisting with a live fetus at 13 weeks of gestation. At 36 weeks of gestation, a cesarean section was performed due to elevated serum human chorionic gonadotropin (hCG) levels, and a male infant with a normal appearance and weighing 2,688 g was delivered. CONCLUSION: If the patient desires to try to carry the fetus to viability after counseling on the possible associated risks of malignancy, it is possible to achieve fetal viability if (1) there is decline in the serum hCG level after it peaks before the second trimester, (2) ultrasound reveals degeneration of the molar part, and (3) there are no complications of pregnancy.     

Placental pathology casebook. Complete hydatidiform mole with coexistent term twin pregnancy.

A case of twin pregnancy consisting of a complete hydatidiform mole with a coexistent, viable fetus is presented. The case is distinctive for its presentation on ultrasound, its unusually low levels of serum hCG, its remarkable histology, and its term delivery.     

Genetically identified complete hydatidiform mole coexisting with a live twin fetus: comparison with conventional diagnosis

Twin pregnancy consisting of a complete hydatidiform mole (CHM) along with a live co-existing fetus is a rare entity and difficult to diagnose. A 37-year-old Japanese woman demonstrated a living fetus, a placenta and a multicystic mass within one gestational sac on ultrasound at 10 weeks. Termination of the pregnancy was performed, and the specimen was classified as partial mole by macro- and microscopic findings. The karyotype of the molar tissue was 46XX. DNA polymorphism analysis demonstrated that fetal DNA showed bi-parental origin while molar DNA showed paternal origin only. Thus, this case was erroneously classified by ultrasonography, macroscopic and pathologic findings, then correctly diagnosed as a twin pregnancy with a CHM and co-existing normal twin fetus by DNA polymorphism analysis. Immunohistochemistry of p57(KIP2), the paternally imprinted and maternally expressed gene, supported the genetic diagnosis. This case suggested that conventional diagnostic methods were inadequate for accurate diagnosis of CHM with a co-existing fetus. DNA polymorphism analysis should be requested for the diagnosis of hydatidiform mole, especially in cases where it is difficult to discriminate between partial hydatidiform mole and CHM with a co-existing fetus.     

Twin pregnancy with a complete hydatidiform mole and surviving co-existent fetus

INTRODUCTION: Twin pregnancy with complete hydatidiform mole and co-existent fetus (CHMF) resulting in a healthy take-home baby is rare, with only 30 cases documented in detail in the literature. CASE REPORT: A 29-year-old woman conceived following two cycles of ovulation induction with clomiphene citrate. Successive ultrasound examinations demonstrated a normally growing live fetus alongside a normal placenta and an additional intrauterine echogenic mass with features of hydatidiform mole. At 17 week gestation, serum beta-hCG level was 25.38 multiples of the median. Genetic amniocentesis at 18.5 week gestation showed normal fetal 46XX karyotype. A cesarean section performed at 28 week gestation resulted in the delivery of a live normal female infant and two adjoining placentas. One placenta was normal and the other placenta was composed of vesicles of various sizes. Microscopic examination of the abnormal placenta confirmed complete hydatidifrom mole. The baby did well and serial maternal serum beta-hCG levels showed a declining trend and were undetectable by 7 weeks after delivery. CONCLUSION: Continuation of a twin pregnancy with CHMF is an acceptable option. There is, however, an increased risk of developing pre-eclampsia and fetal loss due to miscarriage. The chance of a live term birth is <50% with nearly 33% of the mothers developing persistent gestational trophoblastic disease after delivery. Thus, close surveillance of an ongoing twin pregnancy with CHMF is mandatory to detect potential early signs of maternal and fetal complications.     

Outcome of Pregnancies Occurring before Completion of Human Chorionic Gonadotropin Follow-Up in Patients with Persistent Gestational Trophoblastic Tumor

OBJECTIVE: To determine the outcome of pregnancies occurring before completion of human chorionic gonadotropin follow-up in patients treated with chemotherapy for gestational trophoblastic tumor. METHODS: Retrospective record review of patients with gestational trophoblastic tumor who conceived before standard hCG follow-up was completed during 1973-1998. RESULTS: Forty-three patients treated for gestational trophoblastic tumors conceived before human chorionic gonadotropin follow-up was completed. The antecedent pregnancy was complete mole in 31 (72.1%) and partial mole in 12 (27. 9%) patients. Of the 43 patients, 39 (90.7%) had stage I, 1 had stage II, and 3 had stage III disease. The mean interval from human chorionic gonadotropin remission to new pregnancy was 6.3 months (range 1-11 months). Ten patients underwent elective termination and four patients were lost to follow-up. Of the remaining 29 patients, 22 (75.9%) had term live births, 3 (10.3%) had preterm delivery, 3 had spontaneous abortion, and 1 (3.5%) had a repeat mole. Two cases of fetal anomalies were detected; one was inherited polydactyly and the other was hydronephrosis. One patient developed choriocarcinoma with lung involvement and underwent cesarean section at 28 weeks; a normal fetus was delivered and no choriocarcinoma was detected in the placenta. CONCLUSION: Pregnancies occurring in patients treated for gestational trophoblastic tumor before standard human chorionic gonadotropin follow-up is completed may continue under close clinical surveillance since the majority have a favorable outcome. However, patients should also be advised of the low but important risk of delayed diagnosis in case tumor relapse develops during early subsequent pregnancy.     

Prenatal diagnosis and management of twin pregnancies complicated by a co-existing molar pregnancy

Recent advances in ultrasound and molecular genetics have increased our understanding and hence enhanced the perinatal management of complete and partial hydatidiform mole. By contrast, the management of a twin pregnancy combining a normal pregnancy with a normal fetus and a complete hydatidiform mole (CHM) remains complex and controversial due to conflicting data from different parts of the world. The aim of this review is to analyse the international literature on twin pregnancies that include a mole, present the complications and outcome of pregnancy and to discuss the perinatal management. Management is complicated and women should be counselled about the maternal and fetal complications, and the pregnancy monitored carefully by a perinatal team with experience in high-risk obstetrics and access to neonatal care. The data reviewed here suggest that a woman who decides to continue with the pregnancy including a CHM must be aware that, overall, she only has a one in four chance of live birth and in around 35% of cases she will develop persistent trophoblastic disease (PTD) after delivery. In ongoing pregnancies, there will be, in at least 20% of the cases, an early onset of pre-eclampsia (PET) and a 29% risk of fetal loss due to late miscarriage, intrauterine death and neonatal death. Maternal serum human chorionic gonadotrophin (MShCG) could be useful in predicting outcome in twin pregnancy combining normal pregnancy and CHM, but this needs to be investigated prospectively.     

Twin pregnancy with a complete hydatidiform mole and co-existent live fetus: two case reports and review of the literature

Introduction  The aim of this study was to report the clinical features, management, and outcome of two cases of complete hydatidiform mole with a coexisting viable fetus and to review the literature. Case reports  In this article, we report on the well-documented follow-up of two cases of twin pregnancies with complete hydatidiform mole and a normal fetus. Genetic amniocentesis showed normal fetal karyotype in both of two cases. In the first case, a live male infant was delivered by a cesarean section because of severe maternal bleeding at 29 weeks of gestation. In the second case, termination of pregnancy was performed due to early onset of severe preeclampsia and vaginal hemorrhage. Conclusion  The chances of a live birth have been estimated between 30 and 35% and the risk of persistent trophoblastic disease is similar to singleton molar pregnancies in complete mole with coexisting fetus pregnancy. Therefore, in these pregnancies, expectant management instead of termination of pregnancy can be suggested.     

Hydatidiform mole and fetus with normal karyotype: support of a separate entity

Repetitive hydatidiform mole was observed in four pregnancies. The pregnancies presented with heavy bleeding and vomiting, but the post-evacuation courses were uncomplicated, with rapid regression of serum hCG levels. Cytogenetic investigations, analyses of restriction fragment length polymorphisms, and flow cytometry in three pregnancies were consistent with diploid, biparental conception as the origin of fetal tissue and molar and nonmolar villi. In one pregnancy, the analyses of cytogenetic markers suggested the coexistence of two different cell lines of dizygotic, biparental origin, whereas DNA analysis was consistent with a single conception. With incomplete genetic information, a hydatidiform mole with coexistent normal fetus is generally considered to result from dizygous twinning comprising an androgenetic complete mole and a normal conception. In the present gestations, the results based on several techniques applied on numerous samples from different tissues render this possibility unlikely. Some of the contradictions between histologic and cytogenetic classifications of hydatidiform mole may be explained by diploid, biparental partial mole, which seems to constitute a separate subgroup within hydatidiform mole. Following chorionic villus sampling or amniocentesis, continued pregnancy may be considered, depending on prenatal diagnosis including genetic marker analysis.     

A case of twin pregnancy with complete hydatidiform mole and coexisting fetus following IVF-ET

Twin pregnancy consisting of complete hydatidiform mole (H-mole) and a coexisting fetus occurs with an estimated incidence of 1 per 22,000–100,000 pregnancies. The incidence of this unusual twin pregnancy with complete H-mole and a coexisting fetus after in vitro fertilization and embryo transfer (IVF–ET) is not thought to be greater than that of general population. We present an unusual twin pregnancy with complete H-mole and a coexisting fetus that occurred following IVF–ET, which was terminated at 21 weeks of gestation and developed into nonmetastatic gestational trophoblastic tumor.     

Obstetrical and oncological outcomes of twin pregnancies with hydatidiform mole and coexisting fetus

ObjectiveTo evaluate the obstetrical and oncological progression of twin pregnancies with hydatidiform mole coexisting fetus (HMCF).Materials and methodsUsing a retrospective method based on patients from the Women's Hospital, Zhejiang University School of Medicine database between January 1990 and October 2020, 17 patients were histologically confirmed as having HMCF, and the patients' prenatal diagnosis, outcomes and development of gestational trophoblastic neoplasia (GTN) were reviewed.ResultsAmong these 17 cases, 11 (64.71%) cases were complete hydatidiform mole coexisting fetus (CHMCF), and 6 (35.29%) cases were partial hydatidiform mole coexisting fetus (PHMCF). The gestational age at diagnosis of CHMCF was significantly earlier than that of PHMCF [9 (8–24) vs. 18 (11–32) weeks, respectively, P < 0.05]. The live birth rate of PHMCF was slightly higher than that of CHMCF (33.33%; 18.18%), but this difference was not statistically significant. The overall rate of GTN incidence of HMCF was 47.06% (8/17), and the GTN rates of PHMCF and CHMCF were 33.33% (2/6) and 54.55% (6/11), respectively. There was no significant difference in the GTN rate between patients who chose to continue pregnancy and those who terminated pregnancy before 24 weeks of gestation. The GTN rate of patients with term delivery was not significantly higher than that of preterm delivery.ConclusionIn HMCF cases, the incidence rate of CHMCF was higher than that of PHMCF, and PHMCF is more difficult to diagnose in the early stage. Continuing pregnancy does not increase the risk of GTN compared to terminating pregnancy. In cases of HMCF, when the fetal karyotype is normal and maternal complications are controlled, it is safe to continue the pregnancy and extend it to term.     

Twin mole and viable fetus: The case for misdiagnosis

OBJECTIVE: To report the Sheffield Trophoblast Centre experience of twin molar gestations and review this in the light of international experience. CASE: Thirty patients with possible twin molar gestations were registered from 1986 to 2004 (during which period 7,200 cases of mole were seen). The accuracy of suspected clinical and histologic diagnoses was investigated. RESULTS: In 10 cases twin mole/fetus had been suspected clinically but not confirmed when products of conception were examined. In 3 of these cases the pregnancy had been therapeutically terminated because of clinical (ultrasound) suspicion of coexisting molar pregnancy. In the 19 cases where twin mole/fetus was suspected, central histopathology review was possible in 14 cases. Only 7 were confirmed. In 2 further cases twin molar gestation was diagnosed on specimens referred for central review as partial mole singleton pregnancies. For confirmed cases the pregnancy outcome was term delivery in 5 cases and miscarriage in 4. CONCLUSION: Clinical and histopathologic diagnosis of twin molar pregnancies is inaccurate in many suspected cases; therefore, a second (expert) opinion should be sought. When the diagnosis is accurate, maternal and fetal complications are common. However, in suspected cases the pregnancy may be allowed to proceed, with caution, if the mother wishes.     

Maternal serum unconjugated oestriol and human chorionic gonadotrophin levels in twin pregnancies: implications for screening for Down's syndrome.

OBJECTIVE--To investigate maternal serum unconjugated oestriol (uE3) and human chorionic gonadotrophin (hCG) levels in twin pregnancies and to consider the implications of the results for antenatal screening for Down's syndrome. DESIGN--Measurement of maternal serum uE3 and hCG levels from 15-22 weeks of gestation in twin and singleton pregnancies. Previously available maternal serum alpha-fetoprotein (AFP) levels were also presented. SETTING--Stored serum samples collected from women receiving routine antenatal care in Oxford were used. SUBJECTS--200 women with a twin pregnancy and, for each, three singleton control pregnancies matched for gestational age (same completed week of pregnancy) and duration of storage of the serum sample (same calendar quarter). RESULTS--The median uE3, hCG and AFP levels in the twin pregnancies were respectively, 1.67 (95% CI 1.56-1.79), 1.84 (95% CI 1.64-2.07) and 2.13 (95% CI 1.97-2.31) multiples of the median (MoM) for singleton pregnancies at the same gestational age. The variance of values for the three serum markers (expressed in logarithms), and the correlation coefficients between any two, were similar in the twin and singleton pregnancies. CONCLUSION--In maternal serum screening programmes for Down's syndrome dividing uE3, hCG and AFP MoM values in twin pregnancies by the corresponding medians for twin pregnancies will, in expectation, yield a similar false-positive rate in twin pregnancies as in singleton pregnancies.     

Term delivery of a complete hydatidiform mole with a coexisting living fetus followed by successful treatment of maternal metastatic gestational trophoblastic disease

ObjectiveA twin pregnancy consisting of a complete hydatidiform mole with a coexisting normal fetus is extremely rare with an incidence of 1/22,000 to 1/100,000. The incidence of preterm delivery is high and few pregnancies reach near term with a viable fetus.Case reportA 34-year-old woman presented at 20 weeks of gestation with increased levels of serum beta human chorionic gonadotropin (beta-HCG) at 4.74 multiples of the median (310277.7 mIU/mL). Ultrasonography showed a hydatidiform mole together with a normal fetus. Fetal karyotyping revealed 46XY. The serum beta-HCG levels were followed up throughout the remainder of the pregnancy. A male infant weighting 2260 g and the molar tissue were delivered at 37 weeks of gestation. The karyotype of the molar tissue showed 46XX and the histopathological report confirmed our diagnosis. At 4 months postpartum, metastatic gestational trophoblastic disease of the lung was diagnosed in the mother by a computed tomography scan due to increased beta-HCG levels. The patient received three unsuccessful cycles of methotrexate and folinate. Another four cycles of chemotherapy consisting of etoposide, methotrexate, actinomycin D, cyclophosphamide, and vincristine (EMA-CO) were initiated and the beta-HCG levels returned to normal. There was no evidence of recurrence in the subsequent 5 years of regular follow up.ConclusionA pregnancy with a complete hydatidiform mole and a living cotwin can be a serious threat to the health of both the mother and the fetus. Early diagnosis depends on a combination of detecting an unusually high level of serum beta-HCG and ultrasound examination. We suggest that continuation of the pregnancy may be an acceptable option and that the pregnancy may continue until term if a normal fetal anatomy is assured and maternal complications are under control. Patients require careful postpartum follow up and any recurrent disease should be managed aggressively.     

Hydatidiform mole with coexistent fetus. Cytogenetic features, diagnosis, and management

Hydatidiform mole with coexistent fetus is an unusual entity caused by two distinct types of pregnancy: the first one is a partial hydatidiform mole, while the second is a twin pregnancy in which a mole coexists with a normal fetus. In these two separate genetic entities, the counseling and the mother-fetus prognosis are different. Two cases of mole with coexistent fetus are reported: a partial hydatidiform mole typically tripliod and a partial mole with unusual diploid karyotype. Prenatal diagnosis is remarkable for the evaluation of fetus development related with his karyotype. Triplody excludes all hope of a non-malformed surviving child and termination of pregnancy is desirable, while normal karyotype the possibility of a continuation of pregnancy may be considered.     

Gestación gemelar bicorial biamniótica con un feto vivo y una mola parcial del otro gemelo

Twin pregnancy with one sac containing a live fetus and a second sac containing a partial embryonic mole is an extremely rare entity. A 22-year-old nulliparous woman was admitted to our hospital at 16 weeks’ gestation due to persistent hyperemesis unresponsive to the usual medication. Ultrasound examination revealed a bichorial biamniotic twin pregnancy with one live fetus and another fetus with absent fetal heart rate and a biometry of 12-13 weeks. The placenta of the second fetus showed signs of diffuse molar changes. The diagnosis was made with chorionic villus sampling biopsy. The patient wanted to continue with the pregnancy and was followed-up as a high risk pregnancy. Labor was induced at 38 weeks’ gestation due to preeclampsia. Outcome was favorable and the patient remains asymptomatic.     

Obstetric history and antibody titer in estimating severity of Kell alloimmunization in pregnancy

OBJECTIVE: To evaluate the usefulness of the obstetric history and the maternal serum Kell antibody titer in the management of pregnancies with Kell alloimmunization. METHODS: In a retrospective cohort study of 41 pregnancies complicated by Kell alloimmunization, the obstetric history, divided into presence or absence of a previous Kell-positive child, and Kell antibody titers in the index pregnancy were correlated with the gestational age at the onset of fetal anemia. RESULTS: Women with a previous Kell-positive child had a lower gestational age at the first intrauterine transfusion compared with those without a previous Kell-positive child (P=.01). However, in two of 29 pregnancies in the latter group, severe fetal anemia requiring transfusion was detected before 20 weeks of gestation. In neither group were maternal Kell antibody titers significantly correlated with gestational age at first intrauterine transfusion (P=.62 and P=.72, respectively). In all but two pregnancies (1:2 and 1:4, respectively), antibody titers were at least 1:32 before the first intrauterine transfusion. CONCLUSION: For timely detection of all cases of severe fetal anemia, Kell-alloimmunized pregnancies with a Kell-positive fetus and titers greater than or equal to 1:2 should be closely monitored from 16 to 17 weeks of gestation onward.