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BMC Pregnancy and Childbirth
Published in: BMC Pregnancy and Childbirth 1/2021

Open Access 01-12-2021 | Fluorescence in Situ Hybridization | Case report

When a vesicular placenta meets a live fetus: case report of twin pregnancy with a partial hydatidiform mole

Authors: Minhuan Lin, Jinzhu Chen, Bing Liao, Zhiming He, Shaobin Lin, Yanmin Luo

Published in: BMC Pregnancy and Childbirth | Issue 1/2021

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Abstract

Background

Hydatidiform moles exhibit a distinctive gross appearance of multiple vesicles in the placenta. The advances in cytogenetic technologies have helped uncover novel entities of hydatidiform moles and enabled elaborate diagnoses. However, management of a vesicular placenta with a coexistent live fetus poses a bigger challenge beyond hydatidiform moles.

Case presentation

A 33-year-old woman was referred to our department for suspected hydatidiform mole coexistent with a live fetus at 24 weeks’ gestation. The patient had conceived through double embryo transplantation, and first-trimester ultrasonography displayed a single sac. Mid-trimester imaging findings of normal placenta parenchyma admixed with multiple vesicles and a single amniotic cavity with a fetus led to suspicion of a singleton partial molar pregnancy. After confirmation of a normal diploid by amniocentesis and close surveillance, the patient delivered a healthy neonate. Preliminary microscopic examination of the placenta failed to clarify the diagnosis until fluorescence in situ hybridization showed a majority of XXY sex chromosomes. The patient developed suspected choriocarcinoma and achieved remission for 5 months after chemotherapy, but relapsed with suspected intermediate trophoblastic tumor.

Conclusion

We report a rare case of twin pregnancy comprising a partial mole and a normal fetus that resembled a singleton partial molar pregnancy. Individualized care is important in conditions where a vesicular placenta coexists with a fetus. We strongly recommend ancillary examinations in addition to traditional morphologic assessment in such cases.
Literature
1.
Richardson MV, Hertig AT. New England's first recorded hydatidiform mole; a historical note. N Engl J Med. 1959;260(11):544–5.PubMedCrossRef
2.
Jacobs PA, Hunt PA, Matsuura JS, Wilson CC, Szulman AE. Complete and partial hydatidiform mole in Hawaii: cytogenetics, morphology and epidemiology. Br J Obstet Gynaecol. 1982;89(4):258–66.PubMedCrossRef
3.
Hui P, Buza N, Murphy KM, Ronnett BM. Hydatidiform moles: genetic basis and precision diagnosis. Annu Rev Pathol. 2017;12:449–85.PubMedCrossRef
4.
Lawler SD, Pickthall VJ, Fisher RA, Povey S, Evans MW, Szulman AE. Genetic studies of complete and partial hydatidiform moles. Lancet. 1979;2(8142):580.PubMedCrossRef
5.
Tham BW, Everard JE, Tidy JA, Drew D, Hancock BW. Gestational trophoblastic disease in the Asian population of northern England and North Wales. BJOG. 2003;110(6):555–9.PubMedCrossRef
6.
Colgan TJ, Chang MC, Nanji S, Kolomietz E. A reappraisal of the incidence of placental Hydatidiform mole using selective molecular genotyping. Int J Gynecol Cancer. 2016;26(7):1345–50.PubMedCrossRef
7.
Tidy J, Seckl M, Hancock BW, on behalf of the Royal College of Obstetricians and Gynaecologists. Management of Gestational Trophoblastic Disease: green-top guideline no. 38 - June 2020. BJOG. 2021;128(3):e1–e27.
8.
Moscoso G, Jauniaux E, Hustin J. Placental vascular anomaly with diffuse mesenchymal stem villous hyperplasia. A new clinico-pathological entity? Pathol Res Pract. 1991;187(2–3):324–8.PubMedCrossRef
9.
Guenot C, Kingdom J, De Rham M, Osterheld M, Keating S, Vial Y, et al. Placental mesenchymal dysplasia: an underdiagnosed placental pathology with various clinical outcomes. Eur J Obstet Gynecol Reprod Biol. 2019;234:155–64.PubMedCrossRef
10.
Ohira S, Ookubo N, Tanaka K, Takatsu A, Kobara H, Kikuchi N, et al. Placental mesenchymal dysplasia: chronological observation of placental images during gestation and review of the literature. Gynecol Obstet Investig. 2013;75(4):217–23.CrossRef
11.
McNamara HC, Kane SC, Craig JM, Short RV, Umstad MP. A review of the mechanisms and evidence for typical and atypical twinning. Am J Obstet Gynecol. 2016;214(2):172–91.PubMedCrossRef
12.
Crooij MJ, Van der Harten JJ, Puyenbroek JI, Van Geijn HP, Arts NF. A partial hydatidiform mole, dispersed throughout the placenta, coexisting with a normal living fetus. Case report. Br J Obstet Gynaecol. 1985;92(1):104–6.PubMedCrossRef
13.
Hsieh CC, Hsieh TT, Hsueh C, Kuo DM, Lo LM, Hung TH. Delivery of a severely anaemic fetus after partial molar pregnancy: clinical and ultrasonographic findings. Hum Reprod. 1999;14(4):1122–6.PubMedCrossRef
14.
Baxi LV, Mansukhani M, Thaker HM, Parravicini E. Complete hydatidiform mole and live fetus in a singleton pregnancy with confined placental mosaicism and fetomaternal hemorrhage: a case report. J Reprod Med. 2014;59(11–12):599–602.PubMed
15.
Kawasaki K, Kondoh E, Minamiguchi S, Matsuda F, Higasa K, Fujita K, et al. Live-born diploid fetus complicated with partial molar pregnancy presenting with pre-eclampsia, maternal anemia, and seemingly huge placenta: a rare case of confined placental mosaicism and literature review. J Obstet Gynaecol Res. 2016;42(8):911–7.PubMedCrossRef
16.
Sun CJ, Zhao YP, Yu S, Fan L, Wu QQ, Li GH, et al. Twin pregnancy and partial hydatidiform mole following in vitro fertilization and embryos transfer: a novel case of placental mosaicism. Chin Med J. 2012;125(24):4517–9.PubMed
17.
Gajewska M, Zygula A, Wielgos M, Szewczyk G. Twin pregnancy with a partial hydatidiform mole and a coexistent live fetus. Diagnostic and therapeutic dilemmas. A case report and the review of literature. Ginekol Pol. 2020;91(10):589–94.PubMedCrossRef
18.
Jones WB, Lauersen NH. Hydatidiform mole with coexistent fetus. Am J Obstet Gynecol. 1975;122(3):267–72.PubMedCrossRef
19.
Fishman DA, Padilla LA, Keh P, Cohen L, Frederiksen M, Lurain JR. Management of twin pregnancies consisting of a complete hydatidiform mole and normal fetus. Obstet Gynecol. 1998;91(4):546–50.PubMed
20.
Tasci Y, Dilbaz S, Secilmis O, Dilbaz B, Ozfuttu A, Haberal A. Routine histopathologic analysis of product of conception following first-trimester spontaneous miscarriages. J Obstet Gynaecol Res. 2005;31(6):579–82.PubMedCrossRef
21.
Zilberman Sharon N, Maymon R, Melcer Y, Jauniaux E. Obstetric outcomes of twin pregnancies presenting with a complete hydatidiform mole and coexistent normal fetus: a systematic review and meta-analysis. BJOG. 2020;127(12):1450–7.PubMedCrossRef
22.
Buck Louis GM, Grewal J, Albert PS, Sciscione A, Wing DA, Grobman WA, et al. Racial/ethnic standards for fetal growth: the NICHD fetal growth studies. Am J Obstet Gynecol. 2015;213(4):449.e1–449.e41.CrossRef
23.
24.
Makrydimas G, Sebire NJ, Thornton SE, Zagorianakou N, Lolis D, Fisher RA. Complete hydatidiform mole and normal live birth: a novel case of confined placental mosaicism: case report. Hum Reprod. 2002;17(9):2459–63.PubMedCrossRef
25.
Gupta K, Venkatesan B, Kumaresan M, Chandra T. Early detection by ultrasound of partial Hydatidiform mole with a coexistent live fetus. WMJ. 2015;114(5):208–11.PubMed
26.
Bifulco C, Johnson C, Hao L, Kermalli H, Bell S, Hui P. Genotypic analysis of hydatidiform mole: an accurate and practical method of diagnosis. Am J Surg Pathol. 2008;32(3):445–51.PubMedCrossRef
27.
Nayeri UA, West AB, Grossetta Nardini HK, Copel JA, Sfakianaki AK. Systematic review of sonographic findings of placental mesenchymal dysplasia and subsequent pregnancy outcome. Ultrasound Obstet Gynecol. 2013;41(4):366–74.PubMedCrossRef
28.
29.
Braga A, Obeica B, Werner H, et al. A twin pregnancy with a hydatidiform mole and a coexisting live fetus: prenatal diagnosis, treatment, and follow-up. J Ultrason. 2017;17(71):299–305.PubMedPubMedCentralCrossRef
30.
Ronnett BM. Hydatidiform moles: ancillary techniques to refine diagnosis. Arch Pathol Lab Med. 2018;142(12):1485–502.PubMedCrossRef
31.
Fukunaga M, Katabuchi H, Nagasaka T, Mikami Y, Minamiguchi S, Lage JM. Interobserver and intraobserver variability in the diagnosis of hydatidiform mole. Am J Surg Pathol. 2005;29(7):942–7.PubMedCrossRef
32.
Colpaert RM, Ramseyer AM, Luu T, Quick CM, Frye LT, Magann EF. Diagnosis and Management of Placental Mesenchymal Disease. A review of the literature. Obstet Gynecol Surv. 2019;74(10):611–22.PubMed
33.
Han LM, Grenert JP, Wiita AP, Quinn M, Fujimoto VY, Rabban JT. Prevalence of partial Hydatidiform mole in products of conception from gestations with fetal Triploidy merits reflex genotype testing independent of the morphologic appearance of the chorionic villi. Am J Surg Pathol. 2020;44(6):849–58.PubMedCrossRef
34.
35.
Xing D, Adams E, Huang J, Ronnett BM. Refined diagnosis of hydatidiform moles with p57 immunohistochemistry and molecular genotyping: updated analysis of a prospective series of 2217 cases. Mod Pathol. 2021;34(5):961–82.PubMedCrossRef
36.
Xie Y, Pei X, Dong Y, Wu H, Wu J, Shi H, et al. Single nucleotide polymorphism-based microarray analysis for the diagnosis of hydatidiform moles. Mol Med Rep. 2016;14(1):137–44.PubMedPubMedCentralCrossRef
37.
DeScipio C, Haley L, Beierl K, Pandit AP, Murphy KM, Ronnett BM. Diandric triploid hydatidiform mole with loss of maternal chromosome 11. Am J Surg Pathol. 2011;35(10):1586–91.PubMedCrossRef
38.
Fisher RA, Nucci MR, Thaker HM, Weremowicz S, Genest DR, Castrillon DH. Complete hydatidiform mole retaining a chromosome 11 of maternal origin: molecular genetic analysis of a case. Mod Pathol. 2004;17(9):1155–60.PubMedCrossRef
39.
Monk D. Genomic imprinting in the human placenta. Am J Obstet Gynecol. 2015;213(4 Suppl):S152–62.PubMedCrossRef
40.
Demond H, Anvar Z, Jahromi BN, Sparago A, Verma A, Davari M, et al. A KHDC3L mutation resulting in recurrent hydatidiform mole causes genome-wide DNA methylation loss in oocytes and persistent imprinting defects post-fertilisation. Genome Med. 2019;11(1):84.PubMedPubMedCentralCrossRef
41.
42.
Zeng C, Chen Y, Zhao L, Wan B. Partial Hydatidiform mole and coexistent live fetus: a case report and review of the literature. Open Med (Wars). 2019;14:843–6.CrossRef
43.
De Franciscis P, Schiattarella A, Labriola D, Tammaro C, Messalli EM, La Mantia E, et al. A partial molar pregnancy associated with a fetus with intrauterine growth restriction delivered at 31 weeks: a case report. J Med Case Rep. 2019;13(1):204.PubMedPubMedCentralCrossRef
44.
Loza AJ, Fang YMV. Complete molar pregnancy coexisting with a normal fetus in the third trimester. Am J Obstet Gynecol. 2019;220(6):600–1.PubMedCrossRef
45.
Gabra MG, Gonzalez MG, Bullock HN, Hill MG. Cell-free DNA as an addition to ultrasound for screening of a complete Hydatidiform mole and coexisting Normal fetus pregnancy: a case report. AJP Rep. 2020;10(2):e176–8.PubMedPubMedCentralCrossRef
46.
Wang Y, Qian H, Wang J. Medical termination of a partial hydatidiform mole and coexisting fetus during the second trimester: a case report. Oncol Lett. 2015;10(6):3625–8.PubMedPubMedCentralCrossRef
47.
Niemann I, Sunde L, Petersen LK. Evaluation of the risk of persistent trophoblastic disease after twin pregnancy with diploid hydatidiform mole and coexisting normal fetus. Am J Obstet Gynecol. 2007;197(1):45.e1–5.CrossRef
48.
49.
Metadata
Title
When a vesicular placenta meets a live fetus: case report of twin pregnancy with a partial hydatidiform mole
Authors
Minhuan Lin
Jinzhu Chen
Bing Liao
Zhiming He
Shaobin Lin
Yanmin Luo
Publication date
01-12-2021
Publisher
BioMed Central
Published in
BMC Pregnancy and Childbirth / Issue 1/2021
Electronic ISSN: 1471-2393
DOI
https://doi.org/10.1186/s12884-021-04160-2

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