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CASE REPORT Table of Contents  
Ahead of print publication
Giant Placental Chorioangioma in a Young Patient Causing Adverse Fetal Outcome


 Department of Pathology, UCMS and GTB Hospital, New Delhi, India

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Date of Submission18-Aug-2022
Date of Decision10-Nov-2022
Date of Acceptance10-Nov-2022
Date of Web Publication19-Jan-2023
 

  Abstract 


Chorioangiomas are benign vascular tumors of the placenta originating from chorionic tissue. They are also known as hemangiomas of the placenta. They occur in approximately 0.5%–1% of all pregnancies. Large chorioangiomas are rare and may lead to serious fetal and maternal complications. Here, we are describing a case of giant placental chorioangioma in a 19-year-old young female (G2A1) who presented to us at 39 weeks of gestation with decreased fetal movements. Ultrasound examination revealed an enlarged placenta with a well-defined 9.4 cm × 9.3 cm heteroechoic area with increased vascularity. Cesarean section was performed in view of fetal distress and a female baby weighing 1.6 kg was delivered. The newborn died within 2 weeks due to pulmonary hypoplasia and hemodynamic failure. The diagnosis of chorioangioma was confirmed with histopathology. This case depicts the necessity of early diagnosis, close fetal monitoring, and timely intervention in achieving a favorable pregnancy outcome.

Keywords: Chorioangioma, complications, placenta, pregnancy outcome


How to cite this URL:
Wadhwa S, Dixit S, Sharma S. Giant Placental Chorioangioma in a Young Patient Causing Adverse Fetal Outcome. J Microsc Ultrastruct [Epub ahead of print] [cited 2023 Feb 8]. Available from: https://www.jmau.org/preprintarticle.asp?id=368037





  Introduction Top


Chorioangioma is a benign nontrophoblastic vascular tumor of the placenta which arises from primitive chorionic mesenchyme. Its incidence is about 1% of all pregnancies.[1] Symptomatic chorioangiomas are quite rare with an incidence between 1:3500 and 1:9000 total births worldwide.[2] Usually, they tend to be small. If they increase in size >5 cm, then they may be associated with adverse fetal and maternal complications such as intrauterine fetal growth restriction, hydrops fetalis, fetal anemia, increased perinatal mortality, preterm labor, and antepartum hemorrhage.[1],[3],[4]

We report a rare case of giant placental chorioangioma in a young patient causing an adverse fetal outcome. Informed/written consent was taken from the patient and ethical clearance was obtained from the institutional ethics committee before the commencement of the study.


  Case Report Top


A 19-year-old female (G2A1) at 39 weeks and 6 days of gestation presented with a history of decreased fetal movements since 1 day. There was no history of pain abdomen/leaking per vagina/bleeding per vagina. On general physical examination, her vital signs were within normal limits. No abnormality was detected in routine antenatal investigations. The pregnancy had been uneventful until 32 weeks of gestational age when abdominal ultrasound revealed an intrauterine single live fetus with breech presentation with normal cardiac activity. The placenta was enlarged, homogeneous, posterior upper segment in position with a well-defined 9.4 cm × 9.3 cm heteroechoic area along the anterior surface in the proximal aspect with internal vascularity.

The Amniotic Fluid Index was 7.8. Fetal weight was estimated to be about 1.9 kg. Fetal ascites and cardiomegaly were present with a cardiothoracic ratio of 77%. Biophysical profile was 4/10.

External os was found to be closed on per vaginal examination, the head was felt high and pelvis was adequate. Nonstress test was found to be nonreassuring showing fetal bradycardia. In view of breech presentation and fetal distress, emergency cesarean section was done. A female baby weighing 1.6 kg was delivered. The baby did not cry at birth. Liquor was meconium stained. Resuscitation was done and the baby was shifted to the neonatal intensive care unit (NICU). The baby stayed in NICU for 14 days and expired due to lung hypoplasia.

The placenta with membranes was sent for histopathological examination. Macroscopically, the placenta was enlarged; weighing 700 g and measuring 20 cm × 12 cm × 6 cm, with solid gray-white, cauliflower-like friable growth measuring 18 cm × 10 cm × 6 cm on the maternal surface [Figure 1]. The cut surface of the mass appeared fleshy, congested, red to tan, and was well demarcated from the surrounding parenchyma. The umbilical cord was eccentrically placed, measuring 42 cm in length, and the cut section showed three lumens. Microscopic examination showed a well-circumscribed tumor consisting of numerous dilated and congested blood vessels having prominent endothelial cells with inconspicuous loose stroma. This histological pattern was suggestive of angiomatous type of chorioangioma. There were no areas of degenerative changes such as necrosis, calcification, or hyalinization. No mitotic figures or nuclear atypia were seen. Sections from the adjacent normal placenta and umbilical cord were morphologically unremarkable, and the placenta was appropriate for gestational age. A final diagnosis of chorioangioma, the angiomatous type was given.
Figure 1: (a) Gross appearance of placenta showing solid gray-white, cauliflower-like friable growth on the maternal surface (black arrow). (b) Encapsulated tumor with numerous dilated and congested blood vessels. (H and E, ×40) (Inset: Showing adjacent normal placental tissue). (c and d) Tumor with the proliferation of variable-sized blood vessels along with few large thick-walled vessels (H and E, ×100). (e) Vessels lined by endothelial cells suggestive of angiomatous pattern (H and E, ×200) (f) High power view showing endothelial cells with bland-looking nuclei, admixed with loose stroma (H and E, ×400)

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  Discussion Top


Chorioangioma is the most common nontrophoblastic, benign tumor of the placenta.[1] It was reported for the first time in 1798 by John Clarke.[5] Risk factors which are commonly associated include high altitudes (hypoxia related), increased maternal age, diabetes, hypertension, multiple pregnancies, and predilection for female babies.[3],[6] Our patient did not have any of these risk factors.

Placental chorioangiomas can be diagnosed either antenatally or postnatally. Larger tumors are mostly picked up antenatally. Ultrasound may show well-circumscribed intraplacental mass which may be hypoechoic- or hyperechoic-containing small anechoic spaces.[1],[7]

Chorioangiomas are differentiated from other conditions such as degenerated myoma, blood clot, and placental teratoma with the help of color Doppler imaging which shows greater blood flow within the feeding vessel of the mass.[4]

Usually, they are found at the fetal surface near the insertion of the umbilical cord. In the present case, the tumor was located on the maternal surface.

Three histological patterns have been described by Marchetti: Angiomatous, cellular, and degenerative, of which the angiomatous form is the most common.[8] Angiomatous pattern consists of proliferation of blood vessels in different stages from capillary to cavernous with variable amounts of vascular and fibrous stromal components.[4],[8]

Chorioangiomas need to be differentiated from other villous capillary lesions such as chorangiosis and chorangiomatosis.

Chorioangiomas are nodular and well-circumscribed tumors showing capillary proliferation. These capillaries are surrounded by trophoblastic cells. Normal chorionic villi usually have five capillaries per villus, whereas in chorangiosis, more than 10 capillaries in at least 10 terminal chorionic villi can be seen in >to 10 noninfarcted tissues in at least three low power fields of the placenta. Each capillary is lined by a thin well-demarcated basement membrane in chorangiosis.

Chorangiomatosis is less well defined and has intermediate features between chorioangioma and chorangiosis. The proliferation of small-sized capillaries lined by a layer of pericytes and loose reticulin fibers in a circumferential pattern is a feature of choriangiomatosis.[9],[10]

Smaller tumors (<5 cm) rarely lead to any adverse maternal or fetal complications, whereas tumors more than 5 cm are known to have life-threatening outcomes by acting as peripheral arteriovenous shunts. Maternal complications commonly encountered are preterm labor, preeclampsia, polyhydramnios, placental abruption, and postpartum hemorrhage. Fetal complications such as fetal anemia, thrombocytopenia, intrauterine growth restriction, hydrops fetalis, congestive cardiac failure, congenital anomalies, and fetal demise.[1],[3],[4],[7]

In our case, no maternal complications were identified. Intrauterine growth restriction and hydrops fetalis were detected in the fetus. The baby required invasive ventilation postdelivery and succumbed to death on day 14 of life.

They are generally managed conservatively. Many researchers believe in the use of combined treatment modalities for a favorable pregnancy outcome such as serial intrauterine transfusions and amniodrainage for the fetus and devascularization of the tumor by absolute alcohol injection, endoscopic laser coagulation, or microcoil embolization of the feeding vessels of chorioangioma.[1],[6],[7]


  Conclusion Top


Our case emphasizes the importance of knowing such lesions to prevent unfavorable fetomaternal outcomes. Therefore, regular monitoring of the patient by serial ultrasound and close surveillance by Doppler is the key to early diagnosis and effective management.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient (s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Fan M, Skupski DW. Placental chorioangioma: Literature review. J Perinat Med 2014;42:273-9.  Back to cited text no. 1
    
2.
Norris M. Placental mass – Clinically suspected chorioangioma. Australas J Ultrasound Med 2009;12:30-2.  Back to cited text no. 2
    
3.
Guschmann M, Henrich W, Entezami M, Dudenhausen JW. Chorioangioma – New insights into a well-known problem. I. Results of a clinical and morphological study of 136 cases. J Perinat Med 2003;31:163-9.  Back to cited text no. 3
    
4.
Theresia E, Nurdiati D, Widodo I. Giant placental chorangioma: The first case report in Indonesia. Hum Pathol Case Rep 2021;23:200472.  Back to cited text no. 4
    
5.
Clarke J. Tumor found in the substance of the human placenta. Philos Trans R Soc Lond 1798;88:361-68.  Back to cited text no. 5
    
6.
Kumari RS, Thiruselam S, Arumugam P. Placental chorioangioma – An infrequent cause for polyhydramnios. Indian J Obstet Gynecol Res 2020;7:611-13.  Back to cited text no. 6
    
7.
Kodandapani S, Shreshta A, Ramkumar V, Rao L. Chorioangioma of placenta: A rare placental cause for adverse fetal outcome. Case Rep Obstet Gynecol 2012;2012:913878.  Back to cited text no. 7
    
8.
Marchetti AA. A consideration of certain types of benign tumors of the placenta. Surg Gynecol Obstet 1939;68:733-43.  Back to cited text no. 8
    
9.
Ogino S, Redline RW. Villous capillary lesions of the placenta: Distinctions between chorangioma, chorangiomatosis, and chorangiosis. Hum Pathol 2000;31:945-54.  Back to cited text no. 9
    
10.
Amer HZ, Heller DS. Chorangioma and related vascular lesions of the placenta – A review. Fetal Pediatr Pathol 2010;29:199-206.  Back to cited text no. 10
    

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Correspondence Address:
Sonal Sharma,
Department of Pathology, UCMS and GTB Hospital, New Delhi - 110 095
India
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/jmau.jmau_71_22



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    -  Dixit S
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