Giant placental chorioangioma with favorable outcome: a case report and literature review of literature

Summary

Objective:
To describe a case of prenatal diagnosis of a giant placental chorioangioma with favorable outcome.

Design:
A case report.

Setting:
Gynecology and Obstetrics Service, Federal University of Triângulo Mineiro (UFTM), Uberaba-MG, Brazil.

Case report:
The placental chorioangioma is the most common benign tumor, but the type giant has a small prevalence, ranging from 1:16.000 to 1:50.000 pregnancies. We reported a case of a patient aged 18, pregnant for the first time, who performed a routine obstetric ultrasound was found to have polyhydramnios associated with placental vascular lesions suggestive of chorioangioma also was defined by fetal magnetic resonance imaging and confirmed by pathological examination.

Keywords:
chorioangioma, placental, pregnancy, ultrasound, magnetic resonance imaging

INTRODUCTION

Chorioangioma is the most common benign tumour of the placenta. It is usually characterised by a single and small size, vascular and enclosed, intraplacental lesion. Small chorioangiomas are normally asymptomatic and do not change the course of gestation [11]. Due to its small size, a chorioangioma is not frequently detected during macroscopic examination, with a total estimated prevalence of 1% [2].

Giant chorioangiomas are defined as those with 4–5 cm in diameter. They are rarer and have a prevalence estimated between 1:9000 and 1:50000 pregnancies [11]. This type of chorioangioma is associated with fetal complications, including hyperdynamic circulation, anaemia, polyhydramnios, hydropsia and intrauterine growth restriction [4, 9].

Doppler ultrasonography is extremely useful in the diagnosis of giant chorioangioma and differentiation from other types of placental tumours. Ultrasonography findings show a well-defined and round-shaped lesion, hypoechoic or hyperechoic, generally vascularised, located near the chorionic plate and the umbilical cord insertion site [10].

Due to the high perinatal mortality rates (30%–40%), several therapeutic interventions, with limited success in most cases, have been indicated for the prenatal period [11].

We hereby discuss a case of giant chorioangioma that evolved with formation of polyhydramnios, spontaneous resolution and a favourable evolution during gestation.

CASE REPORT

An 18-year-old patient, with 24 weeks of gestation and negative serology, was hospitalised in the Division of Maternal-Fetal Medicine, Department of Obstetrics and Gynecology, of Federal University of Triângulo Mineiro (UFTM) with a clinical presentation of acute pyelonephritis. The physical and gynaecological examinations did not show any alterations. The patient underwent an obstetric ultrasonography which showed a single foetus, in cephalic presentation, with heart rate of 152 bpm, preserved foetal morphology, biometry without changes and an estimated weight of 687 g. High placenta previa, grade 1 according to Grannum classification, 1.9 cm thickness, with a hypoechoic appearance, vascularised, with defined limits bulging in the middle third and measuring 8.5 × 4.7 cm (Figures 1 and 2). Presence of a three-vessel umbilical cord and an amniotic fluid index (AFI) of 38 mm. The screening for gestational diabetes mellitus and dosage of beta fraction of human chorionic gonadotropin were normal.

The foetal magnetic resonance (MR) showed the presence of a well-defined and lobulated solid mass of polypoid appearance, comprising the middle third of the placenta and measuring 7.2 × 4.8 cm, with discrete and delayed contrast enhancement. There were some bleeding spots in the interior, characterised by signal hyperintensity in T1-weighted sequences. There were no signal intensity changes in adjacent placenta (Figure 3). The foetal echocardiography examination did not show evidence of congenital heart disease.

The patient received prenatal and ultrasonography follow-up in the Division of Maternal-Fetal Medicine, every 15 days. After 35 weeks of gestation, there was a gradual reduction of the volume of amniotic fluid before it normalised. At 38 weeks of gestation, the patient began having lower abdominal contractions and loss of mucus plug and was hospitalised for labour. It was a caesarean delivery due to umbilical cord prolapse, with the birth of a live newborn weighing 3,250 g. The Apgar score was 9 and 10 in the 1st and 5th min, respectively.

The histopathological examination of the placenta confirmed the ultrasonography diagnostic suspicion of giant chorioangioma with interior necrotic areas (Figure 4).

DISCUSSION

This is a case report of a giant chorioangioma with favourable evolution during the prenatal period and spontaneous resolution of polyhydramnios during gestation, with the birth of a full-term newborn without any complications. This is a rare condition and there are few described cases with a totally favourable evolution. The size of the detected tumour was > 4 cm, and the patient was diagnosed during the ultrasonography examination, performed during the second trimester of gestation (20–24 weeks), which is in accordance with the literature.

Fan and Mootabar [3] recently described a case of giant chorioangioma that also had a favourable evolution during gestation. In this case report, during maternal-foetal follow-up, they detected an increase in maximum peak systolic velocity in the middle cerebral artery and an 8–13.5 fetal growth percentile. After birth, the complementary exams confirmed absence of anaemia, but the weight of the newborn was the expected average for gestational age.

Colour Doppler ultrasonography is an important tool for tumour identification. Besides confirming the presence of vascular channels that communicate with foetal circulation, it also helps to differentiate chorioangiomas from other placental solid masses such as degenerating leiomyoma, placental teratoma, incomplete hydatidiform mole, retroplacental hematoma and mesenchymal dysplasia. MR is also useful for the evaluation of chorioangiomas during pregnancy and is very sensitive in the presence of bleeding [3]. In our case, MR helped detecting bleeding areas in the interior of the tumour, later confirmed by an anatomopathological examination.

The assessment and treatment of diagnosed cases during gestation depend on the maturation and foetal and maternal complications [1]. An early diagnosis and a strict follow-up improve the maternal and foetal prognosis. Small tumours are generally checked every 3–4 weeks, whereas larger tumours are checked at 1–2 weeks intervals [3]. Complications and a high foetal death rate are explained by the existence of shunts and bleeding between maternal and foetal circulation, microangiopathic haemolysis, as well as placental insufficiency caused by tumour growth [11]. The shunt between maternal and foetal circulation promotes hyperdynamic blood flow and an increase in glomerular filtrate rate that, associated with fluid transudation caused by the tumour, leads to polyhydramnios [11]. In this case, the presence of polyhydramnios associated with maximum peak systolic velocity in the middle cerebral artery support the hypothesis of the transudation of fluids from the tumour.

Hyperdynamic blood flow and anaemia caused by maternal-foetal bleeding and microangiopathic haemolysis contribute to the development of foetal heart failure. The progressive deterioration of myocardium function may explain the occurrence of hydropsia and consequently foetal death [1]. Intrauterine growth restriction is also another complication found in giant chorioangioma cases. Tumour growth reduces the area of normal placental tissue required for adequate exchange of nutrients, thus contributing to lower than expected foetal growth [5].

Due to the occurrence of complications during the late phases of gestation, it is necessary to ponder the possibility of elective labour induction following foetal maturity and in the presence of adequate neonatal support. On the other hand, most complications arise at the end of the second trimester of gestation, when birth is not the best option due to foetal prematurity. Amniodrainage and intrauterine blood transfusion are possible options in case of polyhydramnios or foetal anaemia, respectively [8]. Because these procedures do not treat the aetiology of foetal complications, new therapeutic options have been described, such as absolute alcohol-induced sclerosis or laser ablation of tumour circulation [6, 7].

In this case, no additional propaedeutic was employed, except for strict follow-up of gestation. The favourable evolution was probably due to the occurrence of infarct areas in the tumour, which in turn reduced the maternal-foetal shunt and heart and renal hyperflow. Therefore, it was not necessary to perform invasive foetal procedures.

In summary, giant chorioangioma is a benign placental tumour that can evolve and lead to several foetal repercussions. The use of complementary ultrasonography is essential for early diagnosis and proper prenatal follow-up. Tumour necrosis caused by rapid tumour growth probably contributes to the clinical worsening or improvement of maternal and foetal parameters.

Prof. Edward Araujo Júnior, PhD

Department of ObstetricsPaulista School of Medicine

São Paulo Federal University (EPM-UNIFESP)

Rua Carlos Weber, 956, apt. 113 Visage

05303-000 São Paulo

SP, Brazil

e-mail: araujojred@terra.com.br