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CASE REPORT
Year : 2019  |  Volume : 12  |  Issue : 2  |  Page : 174-176  

Incidental detection of chorangioma with chorangiosis of placenta: A rare case report


Department of Pathology, Dr. D.Y. Patil Medical College, Hospital and Research Center, Dr. D. Y. Patil Vidyapeeth, Pune, Maharashtra, India

Date of Submission04-Apr-2018
Date of Acceptance07-Jun-2018
Date of Web Publication25-Mar-2019

Correspondence Address:
Archana Chirag Buch
B-603 Gold Coast, Ivory Estates, Someshwarwadi, Pune - 411 008, Maharashtra
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/mjdrdypu.mjdrdypu_56_18

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  Abstract 


Chorangioma of the placenta is a rare tumor with a frequency of about 1%, which usually presents as a solitary nodule or, less frequently, as multiple nodules. It is benign vascular tumors of the placenta arising from chorionic tissue. Large chorangioma has adverse effects on both mother and fetus. Chorangiosis is characterized by an increase in number of small-sized vascular channel within chorionic villi. Chorangiosis is considered as a hypoxia-related angiogenesis mainly associated with numerous maternal, fetal, and placental disorders. The case presented to highlight incidental detection of this rare tumor of large size without causing any pregnancy disorders or developmental anomalies of the fetus in a young female.

Keywords: Chorangioma, chorangiosis, placental tumor


How to cite this article:
Ingale YP, Buch AC, Ulhas PN, Kumar H. Incidental detection of chorangioma with chorangiosis of placenta: A rare case report. Med J DY Patil Vidyapeeth 2019;12:174-6

How to cite this URL:
Ingale YP, Buch AC, Ulhas PN, Kumar H. Incidental detection of chorangioma with chorangiosis of placenta: A rare case report. Med J DY Patil Vidyapeeth [serial online] 2019 [cited 2019 Apr 19];12:174-6. Available from: http://www.mjdrdypv.org/text.asp?2019/12/2/174/254776




  Introduction Top


Chorangioma is a rare tumor of the placenta with a frequency of about 1%.[1] Chorangiosis is characterized by hypervascular terminal chorionic villi without stromal hypercellularity with a frequency of about 5%–6% and have been associated with high mortality (42%) and major congenital malformations (39%).[1] Although both are rare entity, the association of chorangioma with chorangiosis is reported in the English literature.[2]

From a histological point of view, chorangiomas can be classified into angiomatous (capillary), cellular, and degenerative types. The capillary type is the most common histological subtype.[2]

Chorangiomas are usually symptomless, often overlooked during routine ultrasound examination. Small chorangiomas are usually not diagnosed and are usually without fetal or maternal complications. They can be discovered sometimes postnatally.[3] Larger masses, especially those more than 5 cm, are more easily diagnosed by ultrasound scan, and they may cause complications.[4] We present a rare case of large chorangioma with chorangiosis which was an incidental finding without any maternal or fetal complications in a young female.


  Case Report Top


A 20-year-old nonhypertensive, nondiabetic female with G2P1A1 admitted at 35 weeks of gestation with breech and footling presentation in active labor. Ultrasound done during 9 and 20 weeks revealed no fetal or placental abnormality. Amniotic fluid was adequate. Labor and delivery were uncomplicated. She delivered a healthy female baby. Placenta was delivered completely and sent for histopathology.

Grossly, placenta weighted 1.4 kg, measured 26 cm × 18 cm × 5.5 cm with centrally attach umbilical cord, and radially distributed allantoic blood vessels. There was a large solitary reddish-brown firm mass on the maternal surface near the umbilical cord measuring 7 cm × 6 cm × 5 cm. Cut surface was grayish-white, firm in consistency [Figure 1]. Rest of the placenta was unremarkable. The umbilical cord was centrally placed with three vessels on cut section. Membranes were unremarkable.
Figure 1: Cut surface of the placenta showing large grayish-white well-circumscribed mass (arrow) on maternal surface

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On microscopy, a neoplasm was seen composed of numerous proliferation of small capillary [Figure 2] with occasional large-sized vascular spaces. Section from the surrounding normal placenta showed chorionic villi with an increased number of capillary-sized vascular channels varying in number from 10 to 12/villi in 10 different regions [Figure 3]. Umbilical cord and membrane were unremarkable. Hence, the final diagnosis of chorangioma with chorangiosis, of placenta was made.
Figure 2: (a) Photomicrograph of chorangioma showing numerous small capillary-sized channels with occasional large-sized vascular spaces (H and E, ×100), (b) higher magnification H and E, ×400)

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Figure 3: Photomicrograph of chorangiosis showing an increased number of capillary-sized channels per villi (H and E, ×100 and Inset H and E, ×400)

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  Discussion Top


Chorangioma is a nontrophoblastic tumor characterized by abnormal vascular development within the placental parenchyma. It is most frequently observed in the third and less frequently in the second trimester of pregnancy as a solitary nodule or less frequently as multiple nodules.[5] It may present as an incidental finding and has no malignant potential. An increased incidence of chorangioma is associated with maternal age, hypertension, diabetes, female sex of the newborn, premature labors, first delivery, and multiple pregnancies.[6] Our case had preterm labor and female newborn as an associated factors.

Chorangioma has no fibrous capsule and sharp demarcation from the surrounding placental parenchyma by a single or less frequently double layer of chorionic epithelium. It is most frequently found on the fetal surface of the placenta, often in the vicinity of umbilical cord insertion, with larger tumors being usually attached to the chorion. In minority of cases, it occurs on the maternal surface where the tumor replaces whole or part of the placental lobe. Our case is rare as it is seen on the maternal surface replacing part of the lobe of the placenta.

It is named as chorangioma as it is a benign angioma arising from the chorionic tissue. These lesions are sometimes classified into placental hamartomas rather than true neoplasia.[7] On gross examination, it is well-circumscribed, with the fleshy congested red-to-tan cut surface.

Three histological patterns of chorangioma have been described by Marchetti:[8] three histological types angiomatous, cellular and degenarative. Angiomatous type is composed of numerous proliferative blood vessels in various stages of differentiation from capillary to cavernous. The amount of vascular and fibrous stromal component can vary. Cellular type shows a predominance of the stromal component with only few ill-formed vessels. Degenerative type shows necrosis calcification, myxoid changes, hyalinization, or fat accumulation. Angiomatous type is the most common which was seen in the present case.

The clinical significance of chorangioma is size dependent. Small chorangiomas possess no clinical significance, while chorangiomas, >5 cm or multiple, may be associated with polyhydramnios, hemorrhage, premature delivery, premature placental separation, and placenta previa.[9] These manifestations may result in severe fetal distress and intrauterine death. They may also lead to nonimmune hydrops fetalis.[9] Anemia, thrombocytopenia, or congestive cardiac failure may be seen in a neonate.[1]

The normal terminal chorionic villi should contain no more than five vascular channels, even when the same vessel is present in more than one plane of section.[10] The diagnostic criteria of chorangiosis were the presence of 10 villi, each with 10 or more vascular channels in 10 or more areas of 3 or more of random, and noninfracted placental areas.[10] Chorangiosis is considered as hypoxia-related angiogenesis mainly associated with numerous maternal, fetal, and placental disorders.[11]

Chorangiosis also has an important potential clinical significance and should be mentioned in the pathology report so that the patient should be investigated for associated conditions such as diabetes, anemia, syphilis, and preeclampsia.

We also observed associated chorioangiosis in the placental villi. This possibly resulted from a compensatory mechanism due to the reduction of the functional parenchyma.

Differential diagnosis of chorangioma includes chorangiosis and chorangiomatosis, which presents a diffuse or more, often a focal proliferation of villous angioblastema with villi that are not present in chorangioma. Chorangiosis has to be differentiated from chorangiomatosis. Chorangiomatosis is seen before 32 weeks of gestation and involves more proximal elements of villous tree, show increased stromal cellularity and stromal collagenization.[11] Chorangiosis is more common after 37 weeks of pregnancy, it is a diffuse process involving the tips of terminal villi and has numerous closely approximating capillaries with the intact basement membrane.[11]

The case was presented to highlight incidental detection of large chorangioma with chorangiosis on the maternal surface of placenta without any maternal and fetal complications

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.
Altshuler G. Chorangiosis. An important placental sign of neonatal morbidity and mortality. Arch Pathol Lab Med 1984;108:71-4.  Back to cited text no. 1
    
2.
Singla S, Kaushik A, Ahluwalia C, Singla G, Manda AK. Chorangioma with chorangiosis, placenta: A rare entity of clinical significance. Ann Pathol Lab Med 2016;3:146-8.  Back to cited text no. 2
    
3.
Bashiri A, Furman B, Erez O, Wiznitzer A, Holcberg G, Mazor M, et al. Twelve cases of placental chorioangioma. Pregnancy outcome and clinical significance. Arch Gynecol Obstet 2002;266:53-5.  Back to cited text no. 3
    
4.
Duro EA, Moussou I. Placental chorioangioma as the cause of non-immunologic hydrops fetalis; a case report. Iran J Pediatr 2011;21:113-5.  Back to cited text no. 4
    
5.
Lež C, Fures R, Hrgovic Z, Belina S, Fajdic J, Münstedt K, et al. Chorangioma placentae. Rare Tumors 2010;2:e67.  Back to cited text no. 5
    
6.
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. 6
    
7.
Kuhnel P. Placental chorioangioma. Acta Obstetr Gynecol Scand 1933;13:143-5.  Back to cited text no. 7
    
8.
Marchetti AA. A consideration of certain types of benign tumors of the placenta. Surg Gynecol Obstetr 1939;68:733-43.  Back to cited text no. 8
    
9.
D'Ercole C, Cravello L, Boubli L, Labit C, Millet V, Potier A, et al. Large chorioangioma associated with hydrops fetalis: Prenatal diagnosis and management. Fetal Diagn Ther 1996;11:357-60.  Back to cited text no. 9
    
10.
De La Ossa MM, Cabello-Inchausti B, Robinson MJ. Placental chorangiosis. Arch Pathol Lab Med 2001;125:1258.  Back to cited text no. 10
    
11.
Caldarella A, Buccoliero AM, Taddei GL. Chorangiosis: Report of three cases and review of the literature. Pathol Res Pract 2003;199:847-50.  Back to cited text no. 11
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]



 

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