UNASSIGNED: Post-molar gestational trophoblastic neoplasia (pGTN) develops in about 15% to 20% of complete hydatidiform mole (CMH). Commonly, pGTN is diagnosed based on hCG monitoring following the molar evacuation. To date, no detailed information is available on how fast can pGTN develop from CHM. However, the concurrence of CHM and pGTN is extremely rare.
UNASSIGNED: A 29-year-old woman presented to the gynecology department with irregular vaginal bleeding and an elevated hCG serum level. Both ultrasound and MRI showed heterogeneous mass in uterine cavity and myometrium. Suction evacuation was performed and histologic examination of the evacuated specimen confirmed complete hydatidiform mole. Repeated ultrasound showed significant enlargement of the myometrium mass one week after the evacuation. pGTN with prognostic score of 4 was then diagnosed and multi-agent chemotherapy regimen implemented with a good prognosis.
UNASSIGNED: In rare cases, CMH can rapidly progress into pGTN. Imaging in combination with hCG surveillance seems to play a vital role guiding timely diagnosis and treatment in the specific condition. Low-risk gestational trophoblastic neoplasia (GTN) should be managed stratified according to the individual situation.

UNASSIGNED: Twin pregnancy combining a complete mole and a normal fetal pregnancy with its own healthy trophoblast is a rare entity. A partial molar pregnancy almost always ends in miscarriage due to a triploid fetus.
METHODS: We report the case of a 43-year-old female patient admitted for bleeding during the 20th week of pregnancy. Pelvic ultrasound showed the combination of a complete hydatidiform mole and a normal fetal pregnancy. The decision to medically terminate the pregnancy was taken after consultation with the family. Examination of the placenta and histological study confirmed the diagnosis of complete hydatidiform mole associated with a normal fetus. The evolution was uneventful.
UNASSIGNED: Twin pregnancy combining a complete mole and a normal fetal pregnancy with its own healthy trophoblast is a rare entity that should not be misdiagnosed. There is still no consensus in terms of therapeutic attitude, the dilemma remains and the decision should always include the couple after a thorough explanation of all the risks.
CONCLUSIONS: Our case reaffirms that to successfully manage this rare yet life-threatening condition, heterotopic pregnancy should be included in the differential diagnosis for any gravid women presenting with persistent abdominal pain, abnormal bleeding and/or extrauterine mass.

Twin pregnancies with complete hydatidiform mole and coexisting live fetus are very rare, with only about 300 reported cases. This type of pregnancy is considered a high obstetric risk due to the possibility of severe maternal-fetal complications. Although the clinical and ultrasound findings can be highly suggestive of this type of pregnancy, the definitive diagnosis is usually reached by histopathological examination. The differential diagnosis usually includes partial hydatidiform mole and hydropic pregnancies, which can present similar findings in specimens from the first trimester of pregnancy and thus it is important to interpret correctly the differentiating features. The use of immunohistochemistry for p57 can prove very useful, although some cases show an aberrant expression. We present a case of a twin pregnancy with complete hydatidiform mole associated with a live fetus, with magnetic resonance imaging and ultrasound for radiopathological correlation. We discuss the differential diagnosis and the utility of p57 immunohistochemistry.

OBJECTIVE: We present two cases of triplet pregnancy with complete hydatidiform mole (CHM) in contrasting outcomes and discuss the complications of mothers and outcomes of fetuses through a literature review, raising an important issue on the management of this special pregnancy.
METHODS: We share our manage experience for two cases of triplet pregnancy with CHM and retrospectively analyze 18 similar pregnancies reported previously with different pregnancy outcomes.
RESULTS: In our cases, one case receiving Clomiphene ovulation induction delivered two live fetuses by cesarean section at 30+ weeks without GTN (gestational trophoblastic neoplasia), unfortunately, the other case following ICSI-ET terminated the pregnancy in the setting of complications at 18+ weeks without GTN. No severe complications were detected during pregnancy and no pGTD was developed after delivery in neither of the pregnant.
CONCLUSIONS: Co-existing complete hydatidiform mole in multiple pregnancies may become more common owing to the spreading use of ART. The decision for whether continue pregnancy depending on the personalized conditions including the complications of the pregnancy, the outcomes of the fetuses, the gestational age for delivery, and the potential progression of persistent gestational trophoblastic disease (pGTD). Furthermore, close monitor is necessary for the pregnant with triplet pregnancy with CHM who want to continue pregnancy.

Distinction of hydatidiform moles (HM) from non-molar (NM) specimens and subclassification of HM as complete hydatidiform mole (CHM) versus partial hydatidiform mole (PHM) are important for clinical practice and investigational studies. The issue of diagnostic reproducibility is still unsolved, the lack of diagnostic accuracy based on morphology is substantial with an important interobserver variability, even between experienced gynecologic pathologists. Many ancillary techniques have been investigated in the last years to refine HM diagnosis. p57 (a paternally imprinted, maternally expressed gene) immunohistochemistry, based on the unique genetics of CHM (purely androgenetic), PHM (diandric triploid), and NM specimens (biparental, with allelic balance) can identify CHMs, which lack p57 expression because of a lack of maternal DNA. However, although its role in HM diagnosis is pivotal, it does not allow the distinction of PHM from NM specimens, both of which express p57 due to the presence of maternal DNA. Molecular genotyping, which compares villous and decidual DNA patterns to determine the parental source and ratios of polymorphic alleles, distinguishes purely androgenetic CHM from diandric triploid PHM, and both of these from NM specimens. Beyond the claim of establishing a \"diagnostic truth\", exceptions and peculiar genetic scenarios in the origin of rare CHM and PHM should be kept in mind when approaching any ancillary technique. An algorithmic approach, even in settings with limited resources, can help the pathologists in the diagnostic dilemma of diagnosis of first trimester abortions.

Epidemiological data on obstetric and oncologic complications in twin pregnancies combining a complete hydatidiform mole (CHM) coexisting with a normal fetus and placenta are limited.
To evaluate perinatal and obstetric outcomes for mother and fetus and risk of gestational trophoblastic neoplasia (GTN) in twin pregnancies including a CHM.
PubMed, MEDLINE and EMBASE and the grey literature were searched for articles published between May 1980 and May 2019 using a protocol designed a priori and registered on PROSPERO (CRD42018112524).
Observational cohort studies of four or more cases confirmed by histopathology and providing data on pregnancy outcomes and GTN.
Two reviewers independently reviewed abstracts and full-text articles. The quality of the studies was assessed with the Newcastle-Ottawa scale and a meta-analysis was performed.
Of the 344 abstracts identified, 14 studies (244 cases) met the eligibility criteria. The incidence of maternal complication in ongoing pregnancies was 80.8% and included vaginal bleeding, hyperthyroidism and pre-eclampsia. There were overall 91 (50%) live births in ongoing pregnancies and 83 (34%) of the total cases were subsequently diagnosed with GTN. Substantial and significant (P < 0.001) heterogeneity was found for the incidence of preeclampsia indicating variability in reporting the incidence of some obstetric complications between studies.
Patients diagnosed with a twin pregnancy combining a CHM and an apparently normal fetus have a high risk of perinatal complications, low live-birth rates and around a third of them will develop a GTN and should be managed by specialised multidisciplinary teams.
Our study indicates a high rate of obstetric and oncologic complications in patients presenting with a complete hydatidiform mole and coexistent normal fetus.

BACKGROUND: Distinguishing hydatidiform moles (HMs) from nonmolar specimens and the subclassification of HM are important because complete hydatidiform mole (CHM) is associated with an increased risk of development of gestational trophoblastic neoplasia. However, diagnosis based solely on morphology has poor inter-observer reproducibility. Recent studies have demonstrated that the use of p57KIP2 immunostaining improves diagnostic accuracy for CHM.
OBJECTIVE: To evaluate the accuracy of p57KIP2 immunostaining compared with molecular genotyping for the diagnosis of CHM.
METHODS: Major databases were searched from inception to March 2017 using the terms \'hydatidiform mole\', \'p57\', and \'genotyping\', with their variations, and the search limit for the relevant study design.
METHODS: Any cross-sectional study, case series, case-control study, cohort study, or clinical trial that evaluated the accuracy of p57KIP2 immunostaining for the diagnosis of CHM compared with genotyping was included. Case reports, narrative reviews, expert opinions, and animal testing were excluded.
METHODS: Extracted accuracy data were tabulated and pooled using a hierarchical bivariate random effects model.
RESULTS: Bivariate meta-analysis produced a summary sensitivity of 0.984 (95% CI: 0.916-1.000) and specificity of 0.625 (95% CI: 0.503-0.736) with significant heterogeneity for specificity (I2 = 71.8, chi-square P = 0.029). The pooled summary diagnostic odds ratio was 56.54 (95% CI: 11.03-289.74) with no heterogeneity (I2 = 0.00%, chi-square P = 0.67). The diagnostic performance of the test was high with an area under the curve of (AUC) 0.980.
CONCLUSIONS: p57KIP2 immunostaining is accurate when diagnosing CHM. It can be used as an adjunct test in a combination algorithmic approach.
UNASSIGNED: A meta-analysis to evaluate the accuracy of p57KIP2 compared with genotyping to diagnose CHM.

Distinguishing hydatidiform moles (HMs) from non-molar specimens and the subclassification of HM are important because complete hydatidiform mole (CHM) is associated with an increased risk of gestational trophoblastic neoplasia. However, diagnosis based solely on morphology has poor interobserver reproducibility. Recent studies have demonstrated that the use of p57KIP2 immunostaining improves diagnostic accuracy for CHM.
We will conduct a systematic review of prospective and retrospective studies to evaluate the accuracy of p57KIP2 immunostaining compared with molecular genotyping for the diagnosis of CHM. A high-sensitivity search strategy will be employed in MEDLINE, EMBASE, LILACS, The Grey Literature Report, OpenGrey, OAIster, and Cochrane CENTRAL. Two reviewers will independently screen all identified references for eligibility and extract data. The methodological quality and bias of the included studies will be assessed according to the Quality Assessment of Diagnostic Accuracy Studies (QUADAS-2) tool, and the overall quality of evidence will be assessed using the Grading of Recommendations Assessment, Development, and Evaluation (GRADE) approach. If a meta-analysis is possible, pooled estimates of sensitivity, specificity, and positive and negative likelihood ratios will be calculated using bivariate random-effects models. Statistical heterogeneity will be evaluated with I 2 statistics and explored through sensitivity analysis.
There is considerable overlap between the histological features of molar and non-molar pregnancies and between complete and partial HMs, which results in significant interobserver variability in the diagnosis of CHM and its mimics. Therefore, molecular techniques are used to correctly diagnosis and treat CHM. However, these molecular diagnostic methods are technically difficult to perform, relatively costly, and unavailable in most pathology laboratories. According to our results, p57KIP2 immunostaining appears to be a practical and accurate adjunct for the diagnosis of CHM and its mimics because this technique is relatively simple, reliable, cost-efficient, and rapid. This systematic review will help to determine whether p57KIP2 immunostaining is an adequate alternative diagnostic test for CHM.
PROSPERO CRD42015024181.

Complete hydatidiform (also referred to as hydatiform) mole with coexisting live fetus is an exceedingly rare event. The fetus usually has a normal karyotype, and approximately 25-40% chance of survival, if pregnancy is allowed to continue until reasonable fetal lung maturity is achieved. However, risk of maternal complications including preeclampsia and subsequent trophoblastic disease are significant. We report a case of a 19-year-old primigravida, at 25 weeks gestation with a complete hydatidiform mole and a coexisting live fetus. She developed severe preeclampsia with uncontrolled hypertension, and pregnancy was terminated by caesarean section, after a short course of dexamethasone to accelerate fetal lung maturity. A morphologically normal live female fetus and placenta were delivered without complications, along with a separate mass of complete mole. The postpartum course was complicated by uterine choriocarcinoma with metastases to lung and left kidney, which responded to chemotherapy. Our case is a rare example of a twin gestation composed of a complete hydatidiform mole with a coexisting live fetus, and illustrates the associated spectrum of maternal complications that mandate close pre- and post-natal surveillance.