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Ahmad S, G R D, Subramanyan A, Amalraj S, M E. A Successful Conservative Management of Twin Gestation of Complete Molar Gestation and Co-Existing Normal Fetus: A Case Report. Cureus 2024; 16:e74275. [PMID: 39717331 PMCID: PMC11664112 DOI: 10.7759/cureus.74275] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 11/22/2024] [Indexed: 12/25/2024] Open
Abstract
Twin pregnancies consisting of one normal fetus and one complete mole are very rare. The main concerning risks associated with the continuation of such pregnancy are hyperthyroidism, theca lutein cysts, preeclampsia, and the development of GTD (gestational trophoblastic disease) spectrum (neoplasia) in the mother, which is due to high human chorionic gonadotropin (HCG) values, and intrauterine death and prematurity in the coexistent normal fetus. We report the successful outcome of conservative management in a healthy mother and baby. A 29-year-old patient was diagnosed with a complete mole and coexisting fetus in the 13th week of pregnancy. The risks of continuing with molar pregnancy were discussed in detail with the patient after informed consent to continue the pregnancy was obtained. With close monitoring, the pregnancy progressed, and she had spontaneous preterm labor and delivered an alive, healthy baby at 34 weeks. Post-delivery, the mother and child had no complications. Follow-up beta HCG dropped to less than 5 IU/mL at six weeks post-delivery. However, close follow-up was continued for the next six months, ensuring optimum health for the mother.
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Affiliation(s)
- Sadaf Ahmad
- Histopathology, Apollo Cancer Centre, Chennai, IND
| | - Dhivyambigai G R
- Obstetrics and Gynaecology, Apollo Cradle Hospitals, Chennai, IND
| | | | | | - Elamathi M
- Pathology, Apollo Hospitals, Chennai, IND
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Zhong L, Song L, Yin R, Li Q, Wang D. Risk factors for gestational trophoblastic neoplasia development of singleton normal fetus with partial hydatidiform mole pregnancy: A retrospective cohort and literature review. J Obstet Gynaecol Res 2023; 49:479-486. [PMID: 36328803 DOI: 10.1111/jog.15488] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/10/2022] [Revised: 10/12/2022] [Accepted: 10/20/2022] [Indexed: 11/06/2022]
Abstract
INTRODUCTION Singleton normal fetus with partial hydatidiform mole (PHM) pregnancy is a rare phenomenon. No previous reports have investigated the risk factors of gestational trophoblastic neoplasia (GTN) progression following this condition. METHODS We retrospectively enrolled cases of singleton normal fetuses with PHM pregnancies at West China Second University Hospital, Sichuan University, from 2005 to 2017. Other cases were identified from PubMed databases during 1975 to 2021 for the cohort study. Cox proportional hazards models were applied to evaluate risk factors for GTN progression based on the patient's clinical characteristics. RESULTS Overall, 36 cases of singleton normal fetuses with PHM pregnancies were enrolled. After a median follow-up of 4.0 (0.8-12.0) months, nine (25.0%) patients progressed to GTN. Gestational age at pregnancy termination (hazard ratio [HR] 0.88; 95% confidence interval [CI] 0.78-0.99, p = 0.032), hyperthyroidism (HR 5.75; 95% CI, 1.16-28.50, p = 0.032), and reasons for pregnancy termination (medical indications vs. patients' choice; HR 0.25; 95% CI, 0.06-0.99, p = 0.049) were significantly correlated with GTN progression. Area under the receiver operating characteristic curve (AUC) of gestational age at pregnancy termination to predict non-progression to GTN was 0.784 (95% CI, 0.615-0.903, p < 0.001). A clinically significant cutoff value, that is, gestational age of 24 weeks, was determined by comprehensively considering the cutoff values of AUC and clinical significance of gestational age. CONCLUSIONS Compared to gestational age of pregnancy termination <24 weeks, ≥24 weeks was a protective factor for GTN. Therefore, there is enough evidence to continue pregnancy, except for uncontrolled severe complications, without increasing the risk of GTN progression.
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Affiliation(s)
- Lan Zhong
- Department of Obstetrics and Gynecology, West China Second University Hospital, Sichuan University, Chengdu, China.,Key Laboratory of Birth Defects and Related Diseases of Women and Children (Sichuan University), Ministry of Education, Chengdu, China
| | - Liang Song
- Department of Obstetrics and Gynecology, West China Second University Hospital, Sichuan University, Chengdu, China.,Key Laboratory of Birth Defects and Related Diseases of Women and Children (Sichuan University), Ministry of Education, Chengdu, China
| | - Rutie Yin
- Department of Obstetrics and Gynecology, West China Second University Hospital, Sichuan University, Chengdu, China.,Key Laboratory of Birth Defects and Related Diseases of Women and Children (Sichuan University), Ministry of Education, Chengdu, China
| | - Qingli Li
- Department of Obstetrics and Gynecology, West China Second University Hospital, Sichuan University, Chengdu, China.,Key Laboratory of Birth Defects and Related Diseases of Women and Children (Sichuan University), Ministry of Education, Chengdu, China
| | - Danqing Wang
- Department of Obstetrics and Gynecology, West China Second University Hospital, Sichuan University, Chengdu, China.,Key Laboratory of Birth Defects and Related Diseases of Women and Children (Sichuan University), Ministry of Education, Chengdu, China
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Hemida R, Khashaba E, Zalata K. Molar pregnancy with a coexisting living fetus: a case series. BMC Pregnancy Childbirth 2022; 22:681. [PMID: 36057566 PMCID: PMC9440514 DOI: 10.1186/s12884-022-05004-3] [Citation(s) in RCA: 3] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/14/2022] [Accepted: 08/26/2022] [Indexed: 12/05/2022] Open
Abstract
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).
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Affiliation(s)
- Reda Hemida
- Gynecologic Oncology Unit, Department of Obstetrics and Gynecology, Mansoura University, 35111 Elgomhuria street, Mansoura, Egypt.
| | - Eman Khashaba
- Department of Community Medicine, Faculty of Medicine, Mansoura University, Mansoura, Egypt
| | - Khaled Zalata
- Department of Pathology, Faculty of Medicine, Mansoura University, Mansoura, Egypt
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Mangla M, Kaur H, Khoiwal K. Partial mole with coexistent live fetus: A systematic review of case reports. J Turk Ger Gynecol Assoc 2022; 23:83-94. [PMID: 35642357 PMCID: PMC9160997 DOI: 10.4274/jtgga.galenos.2022.2021-9-11] [Citation(s) in RCA: 5] [Impact Index Per Article: 1.7] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/02/2021] [Accepted: 03/20/2022] [Indexed: 12/01/2022] Open
Abstract
Objective Molar pregnancy coexistent with a live fetus can be a diagnostic and therapeutic challenge. With increasing incidence of multiple pregnancies, there has also been an increase in twin pregnancy with one mole in the recent years. The authors discuss the epidemiology, clinical presentation, and prenatal diagnosis and attempt to design a possible management strategy, to help guide the treating physician, in the management of partial mole with live pregnancy, thereby improving maternal and fetal prognosis. Material and Methods Numerous case reports have been published in various journals regarding management of individual cases of partial molar pregnancy coexistent with live fetus (PMCF). Therefore, we conducted a systematic review of all the case reports and short case series in English concerning partial mole with live pregnancy from 1999 to 2019, that is in the last 20 years. Results In total, 44 case reports of PMCF were analyzed. The mean gestational age at diagnosis was 20+6 (range: 10-40) weeks. Less than half (19/44; 43.2%) were asymptomatic at the time of detection and PMCF was detected on routine scan done for fetal well-being or 11-13-week scan. The majority (56.8%) resulted in the birth of a healthy live fetus. Gestational trophoblastic neoplasia developed in 3/44 (6.8%). Conclusion PMCF involves a high risk of bleeding, preterm labour, intrauterine growth restriction and stillbirth. Successful management of such cases needs prenatal diagnosis, antepartum surveillance and post-natal follow-up. An obstetrician, maternal fetal medicine specialist, gynecology oncologist and neonatal intensivist should be involved in the care of such pregnancies.
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Affiliation(s)
- Mishu Mangla
- Department of Obstetrics and Gynaecology, All India Institute of Medical Sciences, Hyderabad, India
| | - Harpreet Kaur
- Department of Obstetrics and Gynecology, All India Institute of Medical Sciences, Bilaspur India
| | - Kavita Khoiwal
- Department of Obstetrics and Gynecology, All India Institute of Medical Sciences, Rishikesk, India
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Huang TC, Chang KC, Chang JY, Tsai YS, Yang YJ, Chang WC, Mo CF, Yu PH, Chiang CT, Lin SP, Kuo PL. Variants in Maternal Effect Genes and Relaxed Imprinting Control in a Special Placental Mesenchymal Dysplasia Case with Mild Trophoblast Hyperplasia. Biomedicines 2021; 9:biomedicines9050544. [PMID: 34068021 PMCID: PMC8152467 DOI: 10.3390/biomedicines9050544] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [Key Words] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/04/2021] [Revised: 04/30/2021] [Accepted: 05/10/2021] [Indexed: 11/16/2022] Open
Abstract
Placental mesenchymal dysplasia (PMD) and partial hydatidiform mole (PHM) placentas share similar characteristics, such as placental overgrowth and grape-like placental tissues. Distinguishing PMD from PHM is critical because the former can result in normal birth, while the latter diagnosis will lead to artificial abortion. Aneuploidy and altered dosage of imprinted gene expression are implicated in the pathogenesis of PHM and also some of the PMD cases. Diandric triploidy is the main cause of PHM, whereas mosaic diploid androgenetic cells in the placental tissue have been associated with the formation of PMD. Here, we report a very special PMD case also presenting with trophoblast hyperplasia phenotype, which is a hallmark of PHM. This PMD placenta has a normal biparental diploid karyotype and is functionally sufficient to support normal fetal growth. We took advantage of this unique case to further dissected the potential common etiology between these two diseases. We show that the differentially methylated region (DMR) at NESP55, a secondary DMR residing in the GNAS locus, is significantly hypermethylated in the PMD placenta. Furthermore, we found heterozygous mutations in NLRP2 and homozygous variants in NLRP7 in the mother’s genome. NLRP2 and NLRP7 are known maternal effect genes, and their mutation in pregnant females affects fetal development. The variants/mutations in both genes have been associated with imprinting defects in mole formation and potentially contributed to the mild abnormal imprinting observed in this case. Finally, we identified heterozygous mutations in the X-linked ATRX gene, a known maternal–zygotic imprinting regulator in the patient. Overall, our study demonstrates that PMD and PHM may share overlapping etiologies with the defective/relaxed dosage control of imprinted genes, representing two extreme ends of a spectrum.
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Affiliation(s)
- Tien-Chi Huang
- Institute of Biotechnology, National Taiwan University, Taipei 106, Taiwan; (T.-C.H.); (J.-Y.C.); (W.-C.C.); (C.-F.M.)
| | - Kung-Chao Chang
- Department of Pathology, National Cheng Kung University Hospital, Tainan 704, Taiwan;
| | - Jen-Yun Chang
- Institute of Biotechnology, National Taiwan University, Taipei 106, Taiwan; (T.-C.H.); (J.-Y.C.); (W.-C.C.); (C.-F.M.)
| | - Yi-Shan Tsai
- Department of Radiology, National Cheng Kung University Hospital, Tainan 704, Taiwan;
| | - Yao-Jong Yang
- Department of Pediatrics, National Cheng Kung University Hospital, Tainan 704, Taiwan;
| | - Wei-Chun Chang
- Institute of Biotechnology, National Taiwan University, Taipei 106, Taiwan; (T.-C.H.); (J.-Y.C.); (W.-C.C.); (C.-F.M.)
| | - Chu-Fan Mo
- Institute of Biotechnology, National Taiwan University, Taipei 106, Taiwan; (T.-C.H.); (J.-Y.C.); (W.-C.C.); (C.-F.M.)
| | - Pei-Hsiu Yu
- Department of Obstetrics and Gynecology, National Cheng Kung University Hospital, College of Medicine, National Cheng Kung University, Tainan 704, Taiwan;
- Department of Obstetrics and Gynecology, Kuo General Hospital, Tainan 700, Taiwan
| | - Chun-Ting Chiang
- Department and Graduated Institute of Forensic Medicine, College of Medicine, National Taiwan University, Taipei 106, Taiwan;
| | - Shau-Ping Lin
- Institute of Biotechnology, National Taiwan University, Taipei 106, Taiwan; (T.-C.H.); (J.-Y.C.); (W.-C.C.); (C.-F.M.)
- Agricultural Biotechnology Research Center, Academia Sinica, Taipei 115, Taiwan
- Research Center for Developmental Biology and Regenerative Medicine, National Taiwan University, Taipei 106, Taiwan
- Center for Systems Biology, National Taiwan University, Taipei 106, Taiwan
- Correspondence: (S.-P.L.); (P.-L.K.)
| | - Pao-Lin Kuo
- Department of Obstetrics and Gynecology, National Cheng Kung University Hospital, College of Medicine, National Cheng Kung University, Tainan 704, Taiwan;
- Correspondence: (S.-P.L.); (P.-L.K.)
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Zhang RQ, Zhang JR, Li SD. Termination of a partial hydatidiform mole and coexisting fetus: A case report. World J Clin Cases 2019; 7:3289-3295. [PMID: 31667181 PMCID: PMC6819279 DOI: 10.12998/wjcc.v7.i20.3289] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 04/04/2019] [Revised: 08/22/2019] [Accepted: 09/09/2019] [Indexed: 02/05/2023] Open
Abstract
BACKGROUND We describe the treatment strategy for a patient who was found to have a partial hydatidiform mole and coexisting fetus (PHMCF) during the second trimester. The patient was a 38-year-old Chinese woman who had become pregnant following in vitro fertilization and embryo transplantation. We wanted to determine the safest therapeutic strategy to terminate the PHMCF during the second trimester.
CASE SUMMARY In this case, we present a patient who was found to have a PHMCF complicated with serious continuous vaginal bleeding and pre-eclampsia during the second trimester. After careful evaluation, the pregnancy was considered to be unsustainable and was terminated via caesarean section (CS). An infant with weak vital signs and a partially cystic placenta measuring 110 mm × 95 mm × 35 mm were delivered by CS. The patient was discharged after 4 d. The serum levels of β-human chorionic gonadotropin decreased to within a normal range 5 wk after the operation, and no evidence of persistent trophoblastic disease or lung metastases was noticed at the 6-mo follow-up.
CONCLUSION CS termination of PHMCF during the second trimester may be a relatively safe therapeutic strategy.
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Affiliation(s)
- Rui-Qing Zhang
- Shanghai General Hospital, School of Medicine, Shanghai Jiao Tong University, Shanghai 200120, China
| | - Jia-Rong Zhang
- Shanghai General Hospital, School of Medicine, Shanghai Jiao Tong University, Shanghai 200120, China
| | - Shuang-Di Li
- Shanghai First Maternity and Infant Hospital, Shanghai 200120, China
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Kawasaki K, Kondoh E, Minamiguchi S, Matsuda F, Higasa K, Fujita K, Mogami H, Chigusa Y, Konishi I. 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; 42:911-7. [DOI: 10.1111/jog.13025] [Citation(s) in RCA: 14] [Impact Index Per Article: 1.6] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/12/2015] [Revised: 02/10/2016] [Accepted: 03/12/2016] [Indexed: 12/16/2022]
Affiliation(s)
- Kaoru Kawasaki
- Department of Gynecology and Obstetrics; Kyoto University; Kyoto Japan
| | - Eiji Kondoh
- Department of Gynecology and Obstetrics; Kyoto University; Kyoto Japan
| | | | | | | | - Kohei Fujita
- Department of Gynecology and Obstetrics; Kyoto University; Kyoto Japan
| | - Haruta Mogami
- Department of Gynecology and Obstetrics; Kyoto University; Kyoto Japan
| | | | - Ikuo Konishi
- Department of Gynecology and Obstetrics; Kyoto University; Kyoto Japan
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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; 10:3625-3628. [PMID: 26788180 DOI: 10.3892/ol.2015.3743] [Citation(s) in RCA: 7] [Impact Index Per Article: 0.7] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/11/2015] [Accepted: 09/04/2015] [Indexed: 12/18/2022] Open
Abstract
Partial hydatidiform mole and coexisting fetus (PHMCF) is a rare condition that presents a dilemma for physicians and the parents of the fetus, particularly when PHMCF is detected during the second trimester of pregnancy. The present study reports a case of PHMCF terminated by induction of labor via administration of Rivanol at 17 weeks. Follow-up measurements of serum β-human chorionic gonadotropin (β-HCG) levels, as well as imaging studies, indicated the presence of persistent trophoblastic disease (PTD) and lung metastases. The patient was therefore admitted for three courses of chemotherapy. Subsequently, the metastases receded and β-HCG levels decreased to within the normal range. The patient demonstrated no disease recurrence for 1 year. Following a review of the relevant literature, to the best of our knowledge, all PHMCF cases terminated by medical induction of labor during the second trimester resulted in the development of PTD and lung metastases. However, three cases of PHMCF that were terminated by caesarean section during the third trimester did not develop PTD or metastases. The present study therefore hypothesized that medical termination may not be a safe therapeutic strategy for the treatment of PHMCF during the second trimester, and that pregnancy should be allowed to continue empirically.
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Affiliation(s)
- Yinfeng Wang
- Department of Gynecology, Women's Hospital, Zhejiang University School of Medicine, Hangzhou, Zhejiang 310006, P.R. China
| | - Honglang Qian
- Department of Gynecology, Women's Hospital, Zhejiang University School of Medicine, Hangzhou, Zhejiang 310006, P.R. China
| | - Jinhua Wang
- Radiology, Women's Hospital, Zhejiang University School of Medicine, Hangzhou, Zhejiang 310006, P.R. China
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A triploid partial mole placenta from paternal isodisomy with a diploid fetus derived from one sperm and one oocyte may have caused angiogenic imbalance leading to preeclampsia-like symptoms at 19 weeks of gestation. Placenta 2013; 34:631-4. [DOI: 10.1016/j.placenta.2013.03.015] [Citation(s) in RCA: 6] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 12/10/2012] [Revised: 03/14/2013] [Accepted: 03/30/2013] [Indexed: 11/19/2022]
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Sánchez-Ferrer ML, Machado-Linde F, Martínez-Espejo Cerezo A, Peñalver Parres C, Ferri B, López-Expósito I, Abad L, Parrilla JJ. Management of a dichorionic twin pregnancy with a normal fetus and an androgenetic diploid complete hydatidiform mole. Fetal Diagn Ther 2012; 33:194-200. [PMID: 22832009 DOI: 10.1159/000338926] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/06/2011] [Accepted: 04/18/2012] [Indexed: 11/19/2022]
Abstract
We describe a rare case of complete hydatidiform mole with twin live fetus (CHMTF) confirmed by histopathology, flow cytometry, and polymerase chain reaction techniques. No malformations were observed, fetal karyotype was normal and β-human chorionic gonadotropin levels were increased (>100,000 IU/ml). Once the patient had been informed of the risks, it was decided to continue the pregnancy, but termination of pregnancy was necessary at week 13 + 5 due to maternal complications consisting of hyperthyroidism, hypertension and vaginal bleeding, followed by persistent trophoblastic disease (PTD). Patients diagnosed with CHMTF should be informed of all known risks, including the considerable risk of PTD, which is similar to - or according to some reports - even higher than that associated with a singleton complete mole and is not increased by continuing pregnancy. Due to the low number of series published, evidence-based clinical management guidelines are lacking.
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Affiliation(s)
- María Luisa Sánchez-Ferrer
- Department of Obstetrics and Gynecology, Hospital Universitario Virgen de la Arrixaca, El Palmar, Spain.
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