Twin pregnancy consisting of a complete hydatidiform mole and co-existent fetus: report of two cases and review of literature

Hydatidiform mole with co-existing live fetus is a rare entity. Two cases are reported. In the first, complete mole with a co-existing live fetus was suspected on ultrasound examination at 16 weeks of gestation. A termination of pregnancy was performed due to early onset of severe preeclampsia and thyrotoxicosis. In the second case, the patient was admitted at 26 weeks of gestation due to preeclampsia. Genetic amniocentesis at 19 weeks of gestation revealed a normal 46 XX karyotype. Ultrasound examination at 21 weeks of gestation demonstrated two cystic lesions in the fetal liver, wide multicystic placenta and polyhydramnious. Following deteriorating severe preeclampsia, a live female infant (730 g) along with a huge placenta (1350 g) was delivered by a cesarean section. Unfortunately, the newborn died after 35 days. Pathological examination in both cases was consistent with a complete mole co-existing with a viable fetus. During a 1 year follow up period, there was no evidence of persistent or metastatic disease in both cases. Review of literature discussing the diagnostic tools, clinical features, management and outcome of pregnancies with complete mole with a co-existing live fetus is presented.     

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.     

Intraplacental smooth muscle tumor. A case report.

BACKGROUND: Chorioangioma is the leading diagnosis for circumscribed placental lesion noted on ultrasound. Other rare intraplacental tumors may have a significant clinical impact on the mother and fetus. CASE: An intraplacental, hypoechoic area consistent with chorioangioma was noted on ultrasound examination at 17 weeks' gestation. The lesion remained stable in size throughout gestation, with no evidence of fetal hydrops or polyhydramnios. At term, a live-born male infant was delivered vaginally, followed by spontaneous expulsion of the placenta. Histologic examination of the placenta revealed an intraplacental, potentially malignant smooth muscle tumor. Six weeks postpartum, total abdominal hysterectomy and pelvic node sampling were performed. No residual disease was found on the final histologic examination. CONCLUSION: Although ultrasound differentiation is difficult, rare nontrophoblastic tumors should be considered in the differential diagnosis of placental masses. To our knowledge, this is the first report of such a lesion in the placenta.     

Ultrasound diagnosis of coexisting molar pregnancy following referral for abnormal serum screen

Complete hydatidiform mole and coexistent fetus is a rare occurrence. We report a case of a patient referred for abnormal serum screen and vaginal bleeding in the second trimester of pregnancy. Ultrasound revealed a cystic and solid mass along the posterior uterine wall with multiple hypoechoic (honeycomb) areas noted, consistent with molar degeneration of the placenta. Separate from this mass was a coexisting viable fetus with normal fetal anatomy and distinct anterior placenta. At delivery, a large cystic mass of placental tissue appeared to be implanted separately from a normal-appearing fetus and placenta. The mass weighed 184 g and contained multiple 1- to 1.5-cm cysts throughout. Microscopic sections revealed hydropic chorionic villi with central cistern formation and nonpolar trophoblastic hyperplasia with atypia, compatible with complete hydatidiform mole. This case illustrates that the diagnosis of coexisting molar pregnancy after referral for abnormal serum screen should be considered, especially if maternal serum beta-human chorionic gonadotropin is high and the patient has vaginal bleeding.     

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.     

羊膜带综合征一例

本文报道了1例羊膜带综合征(amniotic band syndrome,ABS)病例。该例孕妇于孕22周+4胎儿系统超声提示未见明显异常,孕30周时,B超发现羊水过多,胎儿水肿可能,收住南京医科大学第一附属医院治疗。入院后予控制血糖,行羊水减量术,B超动态监测羊水指数,每日胎心监护。孕32周起,孕妇出现胎动进行性减少,胎心监护渐呈正弦波样改变;孕32周+3时因胎儿窘迫行急诊剖宫产终止妊娠,术中发现脐带的胎盘插入部见片状羊膜自该处延续至胎儿左下肢近踝部皮肤,羊膜分离呈多孔状,新生儿左踝部可见明显带状缩窄环,左足水肿明显,确诊ABS。该羊膜带对胎儿左足影响较为直接,脐带的胎盘插入部附近的片状羊膜对胎儿是否存在影响尚无足够证据。该早产儿于出生24 h后因"新生儿窒息"死亡。     

Complete hydatidiform mole coexisting with a live fetus

The co-existence of a hydatidiform mole with a living fetus is a rare phenomenon. The condition is a dilemma with respect to the diagnosis and management of associated maternal (a risk of maternal complications, such as preeclampsia, hyperthyrodism, and a risk of malignancy) and fetal (elevated risk of spontaneous abortion, neonatal thyrotoxicosis) complications. A 27-year-old woman was referred to our hospital with a diagnosis of hydatidiform mole and live fetus. The pregnancy was unremarkable except for the complaints of excessive nausea and vomiting. Successive ultrasound examinations demonstrated a normally growing live fetus (14 weeks) alongside a normal placenta and an additional intrauterine echogenic mass with features of hydatidiform mole. Genetic amniocentesis at 18 weeks' of gestation showed normal diploid fetal karyotype. At 20 weeks of pregnancy, a control prenatal visit revealed intrauterine fetal death. The follow-up period for two years was unremarkable. In the case of a normal fetal karyotype and the absence of serious signs of maternal pathology, waiting until fetal viability is achieved can justifiably be proposed, however there is still a risk of prenatal complications such as intrauterine death.     

Massive subchorial thrombohaematoma (Breus' mole).

Ten placentae with massive subchorionic thrombohaematomas are reported. This lesion, which is composed of maternal clotted blood, separates the chorionic plate of the placenta from the villous chorion and is traversed by the main cotyledonary branches of the fetal circulation. In some instances it dissects into the layers of the chorionic plate. In anatomical terms it is identical to the lesion which has been described in early pregnancy under the title of Breus' mole. Previous reports suggested that the lesion was confined to early pregnancies and was a consequence of fetal death. In our series all ten placentae were from pregnancies of at least 25 weeks' maturity and six were more than 30 weeks. Seven infants were liveborn and three survived the neonatal period. Differential diagnosis is discussed along with aetiology, which is still obscure although earlier theories implicating fetal death may be discounted.     

Twin pregnancy with single fetal death due to triploidy--a case report

The incidence of multiple pregnancies is associated with the increased risk in maternal and fetal complications. Intrauterine death of one twin in the second trimester is a rare obstetric complication. Authors report a case of a twin pregnancy with triploidy of one fetus and no chromosomal anomaly of the other twin in a dichorionic diamniotic twin pregnancy. Amniocentesis at 16th weeks disclosed triploidy of this fetus who died afterwards at 20th week of gestation. The pregnancy was continued with special care of the mother and the alive fetus. The second twin was successfully delivered by cesarean section in the 41st week of pregnancy due to the intrauterine fetal distress.     

Clinical management and diagnostic possibilities in hydatidiform mole with coexistent fetus.

The goal of the current review is to present the figures essential for counseling, when hydatidiform mole and normal fetus occur together. Previous reviews of prognosis and risks when mole and fetus are observed together did not adjust for differences in genetic constitution and thus varying risks for gynecologic and obstetric complications. A literature search from 1903 to 1989 revealed 113 reports of pregnancies with mole and fetus in which there appeared to be no major malformations or cytogenetic abnormalities; 87 of those were intended to continue. This group provides the most appropriate risk figures, when mole and karyotypically normal child are detected by first or second trimester prenatal diagnosis. Fifty-two pregnancies (59.8 per cent) proceeded to the 28th week without spontaneous abortion or interruption of pregnancy. None of the children delivered before week 28 survived. Of the pregnancies continuing beyond this time 69.2 per cent of the children survived, 7.7 per cent were live-born with unknown long-term outcome, 17.3 per cent died neonatally, and 5.8 per cent succumbed before delivery. Persistent trophoblastic disease was reported in 19.2 per cent of pregnancies interrupted at diagnosis, as well as in 9.1 per cent of those intended to continue. Due to advances in prenatal diagnosis, clinicians will be confronted with counseling in pregnancies with mole and fetus more often than expected from the literature. Chorionic villus biopsy or amniocentesis can disclose those triploid gestations without possibility of a surviving child. First trimester ultrasound demonstration of a partially cystic placenta and abnormal high se-hCG values should initiate prenatal diagnosis for evaluation of the fetal karyotype, before deciding whether to abort or continue the pregnancy.     

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.     

Mirror syndrome resulting from metastatic congenital neuroblastoma.

Neuroblastoma is a tumor of the sympathetic ganglia and adrenal medulla that rarely metastasizes to the placenta. A 21-year-old gravida 3, para 1 at 28 weeks' gestation had an incidental finding of a 3.8-cm fetal renal mass on prenatal ultrasound. Within 1 week, the fetus developed hydrops and was delivered for nonreassuring fetal assessment. The mother developed mirror syndrome as manifested by hypertension, oliguria, and edema. The hydropic infant developed pulmonary hypertension, sepsis, and renal failure. On day of life 4, life support was discontinued. Pathological examination of the placenta revealed disseminated small round blue cells consistent with neuroblastoma. Metastasis of congenital neuroblastoma to the placenta is exceedingly rare, and cases discovered prenatally have resulted in significant maternal morbidity and 100% neonatal mortality.     

Diagnosis and outcome of complete hydatidiform mole coexisting with a live twin fetus

OBJECTIVE: The aim of this study was to highlight the outcome of complete hydatidiform mole (CHM) coexisting with a live co-twin. METHODS: We investigated four cases of such pregnancy by ultrasound, pathological, cytogenetic, and molecular techniques. Information on clinical follow-up and outcome was also available. RESULTS: All four pregnancies were spontaneous: two ended with the delivery of a live-born baby, while the other two were terminated because of signs of serious maternal pathology or intrauterine fetal death. The criteria for carrying on with the pregnancy are reviewed. The immediate outcome depends on the maternal criteria of serious pathology and on the likelihood of intrauterine fetal death. The risk of persistent trophoblastic disease (PTD) is the same as in the case of a singleton complete mole and also seems to be correlated with the zygosity mechanism identified by molecular analysis. CONCLUSION: Hydatiform mole with a live co-twin fetus is a rare obstetric occurrence. In the case of a normal fetal karyotype, it is justifiable to await developments in the absence of maternal complications. However, treatment criteria still need improvement and diligent maternal follow-up is always warranted in the postpartum period.     

Complete hydatidiform mole in a triplet pregnancy coexisting two viable fetuses: case report and review of the literature

We report a rare case of a complete hydatidiform mole with two or more coexisting fetuses where both infants survived without complications. A male infant weighing 1258 g and a female infant weighing 880 g were delivered without complications and discharged 95 days after the birth. The analysis of DNA microsatellite polymorphisms indicated that the mole was of paternal origin and probably homozygous. The mother suffered from multiple pulmonary metastasis of the hydatidiform mole which was detected 3 days after the surgery and was successfully treated with methotrexate. A complete hydatidiform mole with two or more coexisting fetuses produces a dilemma between immediate termination and pregnancy continuation. Although the present case resulted in a favorable outcome, a review of the 14 reported cases suggests that the high fetal loss rate (90%) must be a consideration in the decision regarding management of such a pregnancy.     

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.     

Complete mole coexistent with a twin fetus

We report two cases of a complete hydatidiform mole coexistent with a live fetus diagnosed by DNA polymorphism analysis. A 27-year-old woman revealed symptoms of pre-eclampsia and ultrasound showed multicystic tumor and placenta coexistent with a live fetus at 16 weeks' gestation. The placenta with partly hydropic change and the fetus without anomaly were consequently evacuated. Another 30-year-old woman had a multicystic mass attached to a normal placenta with a 20-week live fetus on ultrasound examination. A hysterotomy was carried out because of persistent bleeding due to placenta previa. In both cases, DNA was extracted from the placental tissue and the tumor, as well as from maternal and paternal blood. Genetic analysis demonstrated that the placental tumor consisted of only paternal origin, which is consistent with the diagnosis of complete hydatidiform mole.     

孕期人乳头状瘤病毒亚临床感染的母婴传播及对胎婴儿的影响

目的　探讨孕中期、孕晚期人乳头状瘤病毒亚临床感染的母婴垂直传播情况及对胎婴儿的影响。方法　采用多聚酶链反应技术对 42例孕中期、32例孕晚期妇女的宫颈分泌物、母血、胎盘、羊水、脐血、新生儿咽部分泌物标本进行HPV - 6、11、16、18型DNA检测。结果　孕中期 :12例宫颈分泌物HPV阳性 (12 / 42 ) ,11例母血HPV阳性 (11/ 42 ) ,9例胎盘组织HPV阳性 (9/ 42 ) ,2 2例羊水标本中 ,仅 1例羊水HPV阳性 (1/ 2 2 ) ;孕晚期 :2 3例宫颈分泌物HPV阳性 (2 3/ 32 ) ,18例母血HPV阳性 (18/ 32 ) ,12例胎盘组织HPV阳性 (12 / 32 ) ,31例羊水及脐血标本中 ,4例羊水HPV阳性 (4/ 31) ,12例脐血HPV阳性 (12 / 31)。 17例异常胎婴儿中有 3例胎儿窘迫、1例新生儿高胆红素血症及 1例无脑畸形儿的脐血或胎盘组织HPV阳性。结论　孕期HPV感染的母婴传播不但可经产道直接接触传播 ,还可经血液、羊水及胎盘发生宫内传播 ,其宫内传播率的高低主要与母血的HPV感染相关。新生儿脐血的HPV感染主要与母血的HPV感染相关 ,而产时新生儿咽部的HPV感染主要与宫颈分泌物HPV感染相关。妊娠期HPV感染可能与胎儿窘迫、新生儿高胆红素血症、胎儿畸形有关     

妊娠合并乙型肝炎病毒感染孕妇胎儿窘迫发病原因分析

目的：探讨妊娠合并乙型肝炎病毒（HBV）感染孕妇胎儿窘迫的病因、预后及治疗方法。方法：对81例妊娠期HBV表面抗原（HBsAg）、HBVe抗原（HBeAg)、HBV核心抗体（HBcAb)和HBV DNA均阳性,肝功能正常的孕妇及其新生儿（研究组）,85例无肝炎病毒感染,肝功能正常的孕妇及新生儿（对照组）的临床资料、血清学检查结果、胎盘病理检查结果和胎儿预后进行分析,并对研究组中76例婴儿在出生后0、1、6月龄时分别注射酵母菌重组乙型肝炎疫苗10μg,24月龄时检测婴儿HBV表面抗体（HBsAb),以评价母婴HBV阻断效果。结果：（1）研究组胎儿窘迫的发生率为38．3％,对照组为16．5％,两组比较差异有显著性（P＜0．05）。（2）HBV感染胎盘可导致绒毛膜血管病。（3胎儿窘迫者,24月龄时母婴阻断率为78．6％,无胎儿窘迫者母婴HBV阻断率为91．7％,两 者比较,差异有显著性（P＜0．05）。结论：妊娠合并HBV感染,可引起胎盘绒毛膜血管病,致使胎盘功能下降,临床表现为胎儿窘迫、进而导致HBV母婴阻断失败。     

A rare cause of perinatal asphyxia: maternal carbon monoxide poisoning

Carbon monoxide (CO) intoxication has serious adverse effects to the mother and fetus and a result of intrauterine hypoxia, it leads to fetal death or severe neurological sequelae. In this article, a preterm infant who was acutely exposed to CO at the 33rd weeks of gestation before delivery was presented. The baby was delivered by emergent cesarean section at the 34th weeks of gestation due to findings of fetal distress and he had severe hypoxic ischemic encephalopathy leading to death. Results and treatment modalities of CO poisoning during pregnancy were reviewed.     

Problems posed by the death of one fetus in a twin pregnancy

Much recent works has highlighted the problems posed by the death of one fetus in utero during multiple pregnancy. This is a rare but serious accident which exposes the mother and the surviving fetus to the transfer of placentary and fetal thromboplastins or to the direct embolization of necrosed fragments of the placenta. Only fetal death occurring during the second and third terms of pregnancy is considered.