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Obstetrics & Gynecology 2003;102:1028-1033
© 2003 by The American College of Obstetricians and Gynecologists
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ORIGINAL RESEARCH

Perinatal Outcome After Prenatal Diagnosis of Placental Chorioangioma

Waldo Sepulveda, MD, Juan L. Alcalde, MD, Carlos Schnapp, MD and Mariana Bravo, CNM

From the Fetal Medicine Center, Department of Obstetrics and Gynecology, Clinica Las Condes; and the Fetal Medicine Unit, San Jose Hospital, University of Santiago de Chile, Santiago, Chile.

Address reprint requests to: Waldo Sepulveda, MD, Queen Charlotte’s and Chelsea Hospital, Hammersmith Hospitals NHS Trust, Centre for Fetal Care, Du Cane Road, London W12 0HS, United Kingdom; E-mail: waldosep{at}hotmail.com.


    ABSTRACT
 TOP
 ABSTRACT
 MATERIALS AND METHODS
 RESULTS
 DISCUSSION
 REFERENCES
 
OBJECTIVE: To review the prenatal complications, management, and perinatal outcome in pregnancies complicated by placental chorioangioma.

METHODS: Cases of placental chorioangioma diagnosed prenatally as part of a prospective, multicentric program for fetal diagnosis and therapy were identified. All cases were evaluated with color flow imaging. In the latter part of the study, three-dimensional power Doppler angiography was also used to study the vascular pattern of the tumor. Information on maternal demographics, prenatal sonographic findings, pregnancy complications, antenatal intervention, and perinatal outcome was obtained by reviewing the medical records or contacting the referring obstetricians.

RESULTS: In the 5-year period from January 1997 to December 2001, 11 cases of placental chorioangioma were diagnosed prenatally. Nine cases were diagnosed in singleton and two in twin pregnancies. Among the nine cases occurring in singletons, five (56%) were associated with pregnancy complications, including polyhydramnios (n = 3), oligohydramnios (n = 2), fetal growth restriction (n = 2), and nonimmune hydrops (n = 1). Amniodrainage was required in one of these cases, allowing prolongation of pregnancy until term. Four (44%) singletons delivered before 35 weeks. Overall, two fetuses died, including one twin due to complications of twin–twin transfusion syndrome and another with hydrops after alcohol injection into the chorioangioma. In four pregnancies, no prenatal complications were detected in spite of continuous growth and vascularity of the placental mass in three of them.

CONCLUSION: Placental chorioangioma is associated with an increased risk of pregnancy complications, the most common being polyhydramnios and preterm delivery. In selected cases, amniodrainage allows continuation of the pregnancy with improving perinatal outcome. Fetuses who develop hydrops are at the highest risk for perinatal death, with limited therapeutic options being available. Close follow-up is advised, even in those cases with no associated findings at the time of the diagnosis.

Chorioangioma is the term used to describe an abnormal proliferation of vessels arising from chorionic tissue.1,2 With a reported incidence of 1%, it represents the most common tumor of the placenta. However, most chorioangiomas are small, asymptomatic lesions that are only found postnatally after careful slicing of the placenta.3 Larger tumors, particularly those measuring more than 4 cm, are rarely seen in obstetric practice but are clinically significant because they are associated with a number of pregnancy complications, including polyhydramnios, nonimmune hydrops, fetal heart failure, fetal anemia and thrombocytopenia, fetal growth restriction, preterm delivery, perinatal death, and maternal preeclampsia.1–3 Although the underlying pathophysiology for these complications has not been fully elucidated, a prominent role for arteriovenous shunting and sequestration of red blood cells and platelets by the tumor has been postulated.1–3

Large chorioangiomas can be readily identified prenatally by sonography as well-circumscribed, solid masses that usually bulge on the fetal surface of the placenta.4,5 They might have similar sonographic appearance to placental hematomas,4 but the use of color Doppler imaging can facilitate the diagnosis if vessels are identified within the tumor.5–9 Most reports on the prenatal diagnosis of chorioangiomas are, however, based on single cases or small series, with significant publication bias toward those associated with the most serious fetomaternal complications.10–16 The aim of this study was to report a large number of cases of placental chorioangioma diagnosed prenatally by sonography and to review the largest series currently available to provide more accurate information on perinatal outcome in pregnancies complicated by this condition.


    MATERIALS AND METHODS
 TOP
 ABSTRACT
 MATERIALS AND METHODS
 RESULTS
 DISCUSSION
 REFERENCES
 
During the 5-year period from January 1997 to December 2001, cases of chorioangioma prenatally detected by sonography as part of a prospective, multicentric program for fetal diagnosis and therapy were identified. The protocol for this study was approved by the local Fetal Medicine Review Board. At referral, the diagnosis of chorioangioma was made by the demonstration of a well-circumscribed mass, with similar or nearly similar echogenicity to the surrounding placental tissue, located either within the placenta or protruding from the fetal surface of the placenta. In all cases, color flow imaging was used to document and quantify blood flow within the mass. The placental mass was measured in its two largest diameters to document the size and growth during pregnancy. In addition, fetal biometry, assessment of amniotic fluid volume, and color Doppler sonography of fetoplacental circulation was performed routinely. Fetal echocardiography was performed in selected cases only. In the latter part of the study, two cases were also evaluated with three-dimensional sonography (Voluson V730, General Electric Medical Systems, Waukesha, WI). In these cases, multiplanar and surface-rendered three-dimensional views were generated, in addition to power Doppler angiography to evaluate the vascular pattern within the chorioangioma.

Pregnancies in which fetomaternal complications developed were managed according to the specific problem and gestational age at presentation. Amniodrainage was considered with an amniotic fluid index of 40 cm or more, a deepest amniotic fluid pocket of 12 cm or more, or in the presence of excessive maternal symptoms.17 At advanced gestations, the decision to deliver the fetus was made on obstetric grounds. In uncomplicated cases, close monitoring, including serial sonograms and non-stress tests, were performed routinely in the third trimester until delivery.


    RESULTS
 TOP
 ABSTRACT
 MATERIALS AND METHODS
 RESULTS
 DISCUSSION
 REFERENCES
 
During the study period, 11 cases of placental chorioangioma were identified including nine in singleton and two in twin pregnancies. Four of these cases were previously reported in a study describing the use of color flow imaging in the differential diagnosis of placental masses.7 Table 1Go displays the relevant clinical and sonographic findings. The mean gestational age at diagnosis was 26 weeks (range, 20–36), the principal reason for referral being the detection of a placental mass in four cases, evaluation of fetal growth in three, and assessment of twin pregnancy in two. The remaining two cases were detected at the routine detailed second-trimester scan.


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Table 1. Prenatal Diagnosis and Management of Placental Choriangioma
 
At referral, four pregnancies were complicated by polyhydramnios, including one with twin–twin transfusion syndrome and one with nonimmune hydrops. Two other fetuses had growth restriction, one with oligohydramnios and the other with normal amniotic fluid volume. The remaining five pregnancies had normal sonographic findings at referral. Prenatal intervention was considered necessary owing to serious pregnancy complications in two cases. In the first (case 1, Table 1Go), tense polyhydramnios and premature contractions at 27 weeks required amniodrainage of 2500 mL. At the time of presentation, the chorioangioma had poor vascular supply confirmed by three-dimensional power Doppler angiography, which remained so after amniodrainage and for the remainder of the pregnancy. After an uneventful prenatal course, the fetus was delivered electively at 37 weeks. Pathologic examination confirmed that the tumor had undergone degenerative changes with partial thrombosis, necrosis, and calcifications. The second (case 2, Table 1Go) was referred at 26 weeks because of progressive nonimmune hydrops and polyhydramnios. At referral the fetus had scalp edema, ascites, cardiomegaly, pericardial effusion, tricuspid regurgitation, abnormal blood flow velocity waveforms in the ductus venosus, and dilated umbilical vein. A highly vascularized placental mass was demonstrated by color flow imaging and three-dimensional power Doppler angiography. In view of the rapid deterioration of fetal condition, the option of chemosclerosis of the chorioangioma with absolute alcohol was offered as an alternative to early preterm delivery and postnatal treatment.18 The procedure was performed percutaneously, but real-time sonography demonstrated transfer of the alcohol into the fetal circulation and acute thrombosis of the umbilical vein leading to persistent bradycardia and fetal death. In three other (cases 3–5, Table 1Go), the gestational age allowed planned delivery before term. In the first, a fetus with polyhydramnios documented from 20 weeks’ gestation onward developed acute oligohydramnios at 33 weeks. In the other two, fetal growth restriction was diagnosed in association with chorioangioma at 32 and 34 weeks. These three fetuses were delivered by cesarean at 33, 34, and 34 weeks, respectively. The former two had an uneventful neonatal course, but the latter developed complications of prematurity requiring admission to the intensive care unit for 25 days.

Two cases of chorioangioma were diagnosed in a twin pregnancy. The first twins were monochorionic–diamniotic and presented with discordant amniotic fluid volume and fetal size at 20 weeks. A small 21 x 11-mm chorioangioma was identified with color flow imaging in relation to the placental area belonging to the larger twin. However, blood flow within the mass was not identified beyond 24 weeks, although continuous growth of the mass was documented, reaching its largest dimension of 45 x 41 mm at 30 weeks. At 28 weeks, the smaller twin presented with absent end-diastolic flow in the umbilical artery and died in utero at 31 weeks, at which time a cesarean delivery was performed. The surviving twin had an uneventful postnatal course and was discharged in good condition on day 36. The second set of twins was dichorionic–diamniotic, and a 28 x 26-mm placental chorioangioma was detected incidentally at 36 weeks during evaluation of fetal growth before induction of labor. The twins were delivered vaginally and weighed 2192 g and 2212 g, respectively. The neonatal course was uncomplicated.

The remaining four pregnancies (cases 6–9, Table 1Go) had uneventful antenatal courses. All these fetuses were delivered at term and had no neonatal complications. Among them, continuous growth and persistent vascularity of the placental mass was demonstrated in three cases. In the other, a 34 x 28-mm intraplacental mass with no flow within it was incidentally detected at 35 weeks during evaluation of fetal growth. The fetus delivered vaginally and weighed 2610 g. Pathology examination confirmed a placental chorioangioma.


    DISCUSSION
 TOP
 ABSTRACT
 MATERIALS AND METHODS
 RESULTS
 DISCUSSION
 REFERENCES
 
This study reports one of the largest series on the prenatal diagnosis and management of placental chorioangioma. Overall, five of the nine cases (56%) detected in singleton pregnancies were associated with fetal complications, including polyhydramnios (n = 3), oligohydramnios (n = 2), fetal growth restriction (n = 2), and nonimmune hydrops (n = 1). In the remaining four, our policy of screening placental masses with color Doppler imaging probably contributed to the diagnosis of otherwise asymptomatic lesions. In addition, a placental chorioangioma was detected in two twin pregnancies, one of which was complicated by twin–twin transfusion syndrome leading to intrauterine death of the donor twin. Most probably, the poor outcome in this case was unrelated to the presence of the placental chorioangioma.

In view of the well-known association between chorioangiomas and poor perinatal outcome,1–3 close antenatal surveillance should be undertaken in all cases, even if no signs of complications are detected at the time of the diagnosis. If complications develop late in pregnancy, planned delivery could be considered, depending on fetal maturity status and local neonatal facilities. However, pregnancies with the most serious complications often present in the late second trimester, at which time delivery is usually not an option. Several prenatal interventions in such cases are currently available. Because the most common complication is polyhydramnios,1–3 amniodrainage of varying amounts of amniotic fluid has been used with excellent results.6,18,19 Intrauterine transfusion in proved cases of fetal anemia has also been reported with variable success rates.11,20,21

Because these procedures are only performed to treat complications and not the cause of the problem, more aggressive approaches aimed to block vascular supply to the tumor have been developed by several investigators. Quintero et al22 performed fetoscopically assisted devascularization of a placental chorioangioma complicated by nonimmune hydrops at 24 weeks. Although the procedure was technically successful, the fetus deteriorated quickly and died 3 days after the intervention. Lau et al23 reported coil embolization of a large, highly vascularized chorioangioma in an anemic, hydropic fetus, using a 20-gauge spinal needle at 24 weeks. The procedure was performed in conjunction with fetal transfusion, but both procedures had to be repeated 1 week later owing to incomplete obliteration of blood flow to the chorioangioma. In spite of a total of 17 pieces of coil inserted and five intrauterine transfusions, the fetus was delivered by emergency cesarean owing to premature labor at 29 weeks and died after delivery. Chemosclerosis with absolute alcohol is another option. Nicolini et al18 first reported this technique in two pregnancies complicated by polyhydramnios and fetal anemia at 24 and 27 weeks, with good perinatal outcome in both cases. Wanapirak et al24 also used this technique to treat a hydropic fetus at 27 weeks. After the procedure, the hydropic changes resolved within a week, although the fetus was delivered at 32 weeks after rupture of membranes and premature labor, but it had an uncomplicated recovery. Jauniaux and Ogle,6 on the other hand, described in their series a fetus with severe hydrops treated with this technique. After a successful procedure, fetal blood sampling was performed but complicated by fetal bradycardia. The fetus had to be delivered by emergency cesarean and died shortly after delivery. We also attempted vascular occlusion by alcohol in one of our cases, which was already complicated by nonimmune hydrops and cardiac failure. There was immediate passage of alcohol to the fetal circulation, which was documented by real-time sonography at the time of the procedure, leading to acute umbilical vein thrombosis and fetal death. The presence of multiple arteriovenous shunts within the chorioangioma and the close proximity of the mass to the cord probably played a critical role for the ablative material to reach the fetal circulation. In this regard, the new three-dimensional power Doppler technology could assist in the prenatal assessment of tumor vascularity, information that might be useful for pregnancy management.

There are several limitations in our study that must be considered, because this probably accounts for important bias in estimating the prevalence and clinical significance of chorioangiomas detected prenatally. First, a significant number of small chorioangiomas could have been missed on gray-scale sonography, because there is no current policy to screen for these frequent lesions of the placenta. Although these might be mostly small and asymptomatic, and therefore of no clinical significance, the possibility that some of them might be associated with isolated polyhydramnios or suboptimal fetal growth is worth considering.25 Second, some chorioangiomas might not have been recognized as such, because blood flow might not be identified within the mass at the time of diagnosis. Indeed, in a previous series of nine proven cases of chorioangioma, three had no blood flow at the time of presentation.6 Similarly, in one of our cases a small chorioangioma was initially identified with color Doppler imaging, but subsequently the blood flow was undetectable, although continuous growth was still present. In two other cases in our series, sonographic findings were highly suspicious of chorioangioma, but no flow was seen within the mass by color flow imaging. Pathologic examination of the placenta after delivery confirmed a placental chorioangioma in both. These cases stress the importance of pathological examination when an "avascular" placental mass is found prenatally and confirms that even large chorioangiomas can present with no blood flow at color Doppler sonography. Third, some lesions might have been discovered in otherwise normal pregnancies and not been referred for evaluation. Because most of the larger series come from tertiary referral centers, there is a strong possibility that lesions not causing perinatal problems were not referred and therefore not analyzed.

To obtain more accurate information on perinatal outcome in pregnancies complicated by chorioangioma, we performed a MEDLINE search of the English-language literature for series reporting more than five cases of placental chorioangioma prenatally diagnosed by sonography. Relevant information regarding perinatal complications and outcome from three large series,6,8,9 including our own data, is presented in Table 2Go. Among the 32 cases reviewed, polyhydramnios was the most common complication (n = 17, 53%) but required amniodrainage in only four cases. Fifteen fetuses (47%) delivered before 35 weeks’ gestation, and fetal growth restriction was present in six cases (19%). There were five cases (16%) of nonimmune hydrops, and seven perinatal deaths or pregnancy terminations. Prenatal treatments attempted in these series included alcohol ablation in two cases complicated with nonimmune hydrops, with perinatal death in both, and amniodrainage in four, with good outcome in all of them.


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Table 2. Placental Chorioangioma: Review of the Literature
 
In summary, our report and the review of previously published series confirm the high rate of perinatal complications associated with placental chorioangioma. It is obvious from this review that there is a significant bias toward publication of large lesions and, therefore, those that are more likely to be associated with perinatal complications. There is also a need for better prenatal interventions because, when severe complications such as nonimmune hydrops arise, the prognosis is dismal, with few therapeutic options available. The use of techniques such as interstitial laser and monopolar thermocoagulation in these cases should be explored.


    Footnotes
 
This work was supported by Sociedad Profesional de Medicina Fetal "Fetalmed" Limitada, Chile.

doi:10.1016/S0029-7844(03)00859-7

Received May 2, 2003. Received in revised form July 9, 2003. Accepted July 17, 2003.


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10. Eldar-Geva T, Hochner-Celnikier D, Ariel I, Ron M, Yagel S. Fetal high output cardiac failure and acute hydramnios caused by large placental chorioangioma. Case report. Br J Obstet Gynaecol 1988;95:1200–3.[Medline]

11. Hirata GI, Masaki DI, O’Toole M, Medearis AL, Platt LD. Color flow mapping and Doppler velocimetry in the diagnosis and management of a placental chorioangioma associated with nonimmune fetal hydrops. Obstet Gynecol 1993;81:850–2.[Abstract/Free Full Text]

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15. Batukan C, Holzgreve W, Danzer E, Bruder E, Hosli I, Tercanli S. Large placental chorioangioma as a cause of sudden intrauterine fetal death. A case report. Fetal Diagn Ther 2001;16:394–7.[Medline]

16. Mara M, Calda P, Zizka Z, Sebron V, Eretova V, Dudor Kinova D, et al. Fetal anemia, thrombocytopenia, dilated umbilical vein, and cardiomegaly due to a voluminous placental chorioangioma. A case report. Fetal Diagn Ther 2002;17:286–92.[Medline]

17. Kyle PM, Fisk NM. Oligohydramnios and polyhydramnios. In: Fisk NM, Moise KJ, eds. Fetal therapy: Invasive and transplacental. Cambridge: Cambridge University Press, 1997:203–26.

18. Nicolini U, Zuliani G, Caravelli E, Fogliani R, Poblete A, Roberts A. Alcohol injection: A new method of treating placental chorioangiomas. Lancet 1999;353:1674–5.

19. As KA, Hagen P, Webb JB, Wijesinghe D. Therapeutic amniodrainage in chorioangioma. J Obstet Gynaecol 1997;17:169–70.[Medline]

20. Hubinont C, Bernard P, Khalil N, Debavche C, Gosseye S, Jauniaux E, et al. Fetal liver hemangioma and chorioangioma: Two unusual cases of severe fetal anemia detected by ultrasonography, and its perinatal management. Ultrasound Obstet Gynecol 1994;4:330–1.[Medline]

21. Haak MC, Oosterhof H, Mouw RJ, Oepkes D, Vandenbussche FP. Pathophysiology and treatment of fetal anemia due to placental chorioangioma. Ultrasound Obstet Gynecol 1999;14:68–70.[Medline]

22. Quintero RA, Reich H, Romero R, Johnson MP, Goncalves L, Evans MI. In utero endoscopic devascularization of a large chorioangioma. Ultrasound Obstet Gynecol 1996;8:48–52.[Medline]

23. Lau TK, Leung TY, Yu SC, To KF, Leung TN. Prenatal treatment of chorioangioma by microcoil embolisation. Br J Obstet Gynaecol 2003;110:70–3.

24. Wanapirak C, Tongsong T, Sirichotiyakul S, Chanprapaph P. Alcoholization: The choice of intrauterine treatment for chorioangioma. J Obstet Gynaecol Res 2002;28: 71–5.[Medline]

25. Mucitelli DR, Charles EZ, Kraus FT. Chorioangiomas of intermediate size and intrauterine growth retardation. Pathol Res Pract 1990;186:455–8.[Medline]




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