CASE REPORT Intisari Sains Medis 2021, Volume 12, Number 1: 68-70 P-ISSN: 2503-3638, E-ISSN: 2089-9084 Chorioangioma in : a case report

Rio Yoga Erlambang1*, I Wayan Artana Putra2 Published by Intisari Sains Medis

ABSTRACT Background: Placental chorioangioma is a rare benign of membrane with 4 cm cervix dilation. Spontaneous non-trophoblastic tumor of the , derived from vaginal delivery was performed and a boy neonate primitive chorionic mesenchyme. It can cause adverse with 4/6 Apgar score and birth weight of 1150 g was outcomes on the mother and fetus, depending on its delivered. Placenta was extracted out completely. There size. Most of the cases are accidentally detected during was no complication after delivery process. Macroscopic ultrasonography because small size tumors are usually and microscopic placental examination confirmed the asymptomatic. However, larger size of tumor can cause initial diagnosis, chorioangioma. 1Resident of Department of Obstetrics and complications such as fetal anemia, fetal hydrops, Conclusion: Early management in determining Gynecology, Faculty of Medicine, Universitas polyhydramnios, premature birth, intrauterine growth the cause of polyhydroamnion is challenging and Udayana, Sanglah General Hospital, Denpasar, Bali, restriction, and fetal death. needs comprehensive investigation. Ultrasonography Indonesia Case report: A 27-year-old primigravida presented is a choice of method in initial detection of 2Obstetrician and Gynecologist, Maternal-Fetal at Sanglah Hospital gynecology polyclinic at 27th-28th polyhydroamnion. Serial ultrasonography is needed to Medicine Division, Department of Obstetrics and week of gestation. She was referred by an obstetric evaluate the amount of amniotic fluid and detect the Gynecology, Faculty of Medicine, Universitas and gynecologist due to polyhydramnion since 26th- probable cause of complications for mother, fetus, and Udayana, Sanglah General Hospital, Denpasar, Bali, th Indonesia 27 week of gestation. A 7.86 x 8.19 cm hypoechoic delivery process. One of the complications is premature cysts with hyperechoic border were found during fetal uterine contraction that causes premature birth. As scanning. Chorioangioma was suspected with SDP 9.11 a result, the fetus may not be viable and this remains *Corresponding author: cm. Amniocentesis was planned, however the patient one of the biggest challenges for clinicians. There are Rio Yoga Erlambang; Resident of Department of Obstetrics and had a premature uterine contraction. By this time, the several possible causes that cannot be ruled out from st nd Gynecology, Faculty of Medicine, Universitas fundal height was accordant with 31 -32 week of polyhydroamnion differential diagnoses, such as a rare Udayana, Sanglah General Hospital, Denpasar, Bali, gestation, and speculum examination showed rupture placental tumor chorioangioma. Indonesia; [email protected] Keywords: preterm, polyhydramnios, chorioangioma. Cite This Article: Erlambang, R.Y., Putra, I.W.A. 2021. Chorioangioma in pregnancy: a case report. Intisari Sains Received: 2020-11-13 Medis 12(1): 68-70. DOI: 10.15562/ism.v12i1.857 Accepted: 2021-03-16 Published: 2021-04-01

INTRODUCTION it can result in adverse mother and been known since the 26th-27th week of neonatal outcomes, such as fetal anemia, gestation when performing antenatal care. Chorioangioma was first described by fetal hydrops, polyhydramnios, premature From fetal scanning, chorioangioma was Clarke in 1978, as the most common 1 birth, intrauterine growth restriction, suspected and esophageal atresia was not tumor of the placenta. Chorioangioma is and fetal death. The fetal and neonatal found. The patient was not experiencing a benign non-trophoblastic tumor of the mortality rate when the tumor size > 5 cm any symptoms. The history of medicine placenta, derived from primitive chorionic is 18%-40%.2-4 Here, we report a rare case during early pregnancy was denied. mesenchyme.2-4 This tumor accounts for 5 of placental chorioangioma who presented From the physical examination, vital approximately 0.6% of all pregnancy. with polyhydramnios. signs were within normal limits. Fundal The clinical significance is related height was 4-finger-width below the to the size of the tumors. Most of the CASE REPORT xiphoid process. The fetus was in cephalic chorioangioma is small in size and often presentation and the fetal heartbeat was A 27-year-old primigravida presented at asymptomatic and accidentally found 156 beats per minute. Fetal biometric USG 1 Sanglah Hospital gynecology polyclinic during routine ultrasonography (USG). showed 27th weeks of gestation, normal at the 27th-28th week of gestation. She was Giant chorioangioma, greater than 5 cm umbilical arterial blood flow (S/D: 2.17; referred by an OBGYN specialist due diameter, is unusual, with incidence rate RI: 0.54; PI: 0.73), increased amniotic fluid ranging from 1/3,500 to 1/9,000. However, to polyhydramnios. The condition has

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the fibrous stroma. Tumour was covered by trophoblastic cells. At one side of the tumor, there was a necrotic area and calcified focus. There were no signs of malignancy from the specimen (Figure 2).

DISCUSSION Placental chorioangioma is the most common of the placenta. The estimated incidence of placental chorioangioma is about 1% in microscopically examined . Most chorioangiomas are small, asymptomatic, and generally have no clinical significance. Large tumors, particularly those measuring more than 5 cm, are rarely seen in obstetric practice but have an association with maternal and fetal complications.6 Large tumors can result in adverse mother and neonatal outcomes, e.g. fetal anemia, fetal hydrops, polyhydramnios, premature birth, intrauterine growth restriction, and intrauterine fetal death.2,3,5 Figure 1. Ultrasonography showed a 7.86 x 8.19 cm hypoechoic cysts with hyperechoic A 7 x 6 x 5 cm spherical solid mass border was present. In this case, the patient suffered from polyhydramnios and later later, the patient revisited the hospital had a premature birth. Polyhydramnios and was hospitalized due to premature is the most common complication of uterine contraction. She complained of chorioangioma, occurring in 14%–28% intermittent abdominal pain and could feel of clinical cases. Three main theories exist fetal movement. Vital signs were normal, to explain this increase in liquor volume. abdomen was enlarged with fundal height Firstly, placental chorioangioma is usually st nd concordant with the 31 -32 gestational located near the umbilical cord insertion, week. Speculum examination showed a and the mechanical compression of rupture of the membrane with 4 cm cervix the umbilical vein by the tumor could dilation. Ultrasonography showed normal obstruct the umbilical vein leading to fetal growth by gestational age, and no greater transudation of fluid. Secondly, structural defect nor fetal hydrops. The excess amniotic fluid may result from a amniotic fluid index was 8.42 cm. The fetal fluid imbalance caused by increased placenta is attached to the back of the production of fetal urine or by congestive uterus. heart failure. The third hypothesis Spontaneous vaginal delivery was postulates that the excess of amniotic fluid performed. A baby boy was born with a results from a transudate through the wall birth weight of 1150 g, with an Apgar score of the abnormal vessels of the tumor, and st th was 4/6 at 1 and 5 minute, respectively. subsequently, through the fetal plate of Figure 2. Placental mass Placenta was extracted out completely, 190 the placenta. Preterm labor mainly caused grams in weight, and 20 x 16 x 16 cm in over-distension and polyhydramnios. size. The three-vessel umbilical cord with The preterm delivery mechanism may (SDP: 13 cm). A 7.86 x 8.19 cm hypoechoic 12 cm in length and 0.8 cm in diameter be partly due to biochemical changes in cysts with a hyperechoic border was also was eccentrically placed. A single 7 x 6 x amniotic fluid, including elevated level.7,8 revealed (Figure 1). 5 cm spherical solid mass was extending An increased incidence rate of The patient was diagnosed with from the maternal surface to the fetal chorioangioma is associated with maternal G1P000 with 27-week-5-day gestation surface. Microscopic examination of the age, hypertension, diabetes, female sex week, single alive, polyhydramnios, placental tissue revealed a well-defined of the newborn, premature labors, first suspected chorioangioma. Amniocentesis tumor, growing from chorionic villi, delivery, and multiple .4,6,9 The (karyotyping) was planned. Six days containing capillary hyperplasia within patient has two of these factors: premature

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labors and first delivery. complications, treatment can be thought IWAP is responsible for supervision, and Doppler ultrasound examination is the of. Amniodrainage of the excessive writing original draft. All author had gold standard in the diagnosis of placental amniotic fluid is an option in the case of reviewed and agree for the final version of chorioangioma during pregnancy. polyhydramnios. In a uterine transfusion, the manuscript. Doppler sonography is the main tool fetoscopy, and laser coagulation of the in the differential diagnosis between vascular shunts are the other management REFERENCES angioma and placental hematoma, options. The proper timing of the labor is 1. Williams Obstetrics. Implantation, teratoma, and myoma uteri. Ultrasound controversial and primarily depends on Embryogenesis, and Placental Development: findings of chorioangioma commonly the fetomaternal complications. Unless Introduction. 24th ed. 2014. pp. 116-125. reveal hypo-or well-circumscribed these complications occur, the patients 2. Arulkumaran S, Sivanesaratnam V, Chatterjee A, Kumar P. Essentials of Obstetrics. 3rd ed. distinctly placenta. This contains cystic should be monitored closely, at least Jaypee Brothers Medical Publishers (P) Ltd. and may or may not contain fibrous septa, every month for small tumors, and every 2004. pp. 82-85. which can create the appearance of a 1-2 weeks for the greater ones.2,3,6 In the 3. Liu CC, Pretorius DH, Scioscia AL, Hull complex mass. Chorangiomas are found case, the amnioreduction procedures AD. Sonographic prenatal diagnosis of on the fetal surface of the placenta around were planned. However, the patient had a marginal placental cord insertion: Clinical importance. Journal of Ultrasound in Medicine. the umbilical cord insertion. Great blood preterm contraction before the procedure 2002;21:627-632. flow in the placental mass and the lacunar was done. Spontaneous vaginal delivery 4. Rhone SA, Magee F, Remple V. The association anechoic areas of the mass or a gross was performed. of placental abnormalities with maternal and feeding vessel within the placental mass neonatal clinical findings: A retrospective CONCLUSION cohort study. Journal of Obstetrics and favors the diagnosis of chorioangioma. Gynaecology Canada. 2003;25(2):123-128. Placental teratomas are cystic or solid Chorioangioma is a rare but challenging 5. DC Dutta’s Textbook of Obstetrics. The Placenta masses with calcifications on sonographic and Fetal Membranes. 7th ed. 2018. pp. 28-37. condition. Antenatal diagnosis of such examination. Teratomas do not have a rich 6. Guschmann M, Henrich W, Dudenhausen cases is a must to prevent and manage vascular supply. In contrast to angiomas, JW. Chorioangiomas-new insights into a well- complications associated with it. Giant known problem. II. An immunohistochemical the echogenicity of hematomas differs placental chorioangiomas are associated investigation of 136 cases. Journal of Perinatal from time to time as the coagulation with an increased risk of fetal demise. Medicine. 2003;31:170-175. process occurs. MRI can be used as an 7. Fan M, Skupski DW. Placental chorioangioma: a Therefore, we emphasize the need to adjunct in cases where the USG diagnosis literature review. Journal of Perinatal Medicine. consider the diagnosis of chorioangioma 2014;42(3):273-279. is equivocal.1,2,5 In this presented case, a with all clinical presentations of 8. Ropacka-Lesiak M, Gruca-Stryjak K, 7.86 x 8.19 cm hypoechoic cysts with a polyhydramnios. Breborowicz G. Nontrophoblastic placental hyperechoic border with normal blood tumors. Neuro Endocrinol Lett. 2012;33:375-9. 9. Kodandapani S, Shreshta A, Ramkumar V, Rao flow was also revealed. CONFLICT OF INTEREST Histologically, three different L. Chorioangioma of Placenta: A Rare Placental Cause for Adverse Fetal Outcome. Case Reports forms may be seen in chorioangiomas: The author declares there is no conflict of in Obstetrics and Gynecology. 2012:913878. angiomatous, cellular, and degenerative interest regarding publication of this study. 10. Marchetti AA. A consideration of certain types forms. The angiomatous form is the most of benign tumors of the placenta. Surgery, common.10 As in this case, the tumor ETHICS IN PUBLICATION Gynecology & Obstetrics. 1939;68:733-743. microscopically showed an angiomatous All patient had received signed informed structure with areas of capillaries, and consent prior to any data collection blood vessels surrounded by placental regarding publication of their medical stroma. data in medical journal. Management of chorioangioma is usually conservative. Fetal treatment AUTHOR CONTRIBUTION of the chorioangioma is controversial. Small tumors can be managed RYE was responsible for data gathering, expectantly. However, for some visualization, and writing original draft.

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