Giant placental chorioangioma: A rare case report.

INTRODUCTION AND IMPORTANCE: Chorioangioma is benign, non-trophoblastic vascular neoplasms of the placenta with an estimated incidence of 1 %. It originates from placental blood vessels. Giant chorioangiomas, larger than 4 cm in diameter, are rare with an incidence ranging between 1/3500 and 1/9000 pregnancies. Giant chorioangiomas easily detected by prenatal ultrasound and are associated with a series of pregnancy and fetal complications. CASE PRESENTATION: A 34-year-old multigravida woman, with twin pregnancy presented with notation of reduced fetal movement. On sonographic examination, first fetus was Intrauterine Growth Restriction (IUGR), stage 1 and the second fetus was small-for gestational age and a well-defined, hypoechoic lesion with increased vascularity measuring 5.8 × 4.7 × 2.5 cm on the fetal surface of the placenta was seen. However, at 35 + 3 weeks, the patient presented with pain in the lower abdomen. Maternal vital signs were within normal ranges. On Physician team discussion, cesarean section was performed. Two female neonates weighing 2260 g and 2400 g were delivered, with normal APGAR scores and physical examinations. And physical examination. The placenta was sent to pathology laboratory. In histopathology numerous proliferative blood vessels was found that confirm with immunohistochemical analysis. Finally, the patient was diagnosed with placental chorioangioma. DISCUSSION: Placental chorioangioma is a rare anomaly in villous capillary development, with uncertain pathogenesis. It is often linked to twin pregnancies, gestational diabetes, maternal hypertension, and female fetal sex. Prenatal sonographic scans are valuable for its identification, revealing a hypoechoic, highly vascular mass confirmed via Doppler ultrasound. In contrast, chorangiocarcinoma, a malignant placental tumor, comprises chorioangioma and proliferating trophoblast cells with distinct histological features. Close monitoring and sonographic evaluations are vital during pregnancy when managing chorioangioma, especially giant ones, known to cause various fetal and pregnancy complications. The decision-making process for delivery in cases of giant chorioangioma should consider fetal complications and gestational age. While some interventions like laser ablation are available, the challenging nature of the patient's response may warrant conservative management in certain instances. CONCLUSION: We report a rare case of giant placental chorioangioma in a 34-year-old twin pregnant patient. Chorioangioma benign vascular neoplasms of the placenta may cause pregnancy and fetal complications.

Large placental chorioangioma with maternal and perinatal morbidity.

INTRODUCTION: Placental chorioangioma is a benign placenta tumour. Majority of cases, the placental chorioangioma are small and no maternal and fetal complications. We highlight a case diagnosed with large placental chorioangioma with an intrapartum event associated with significant maternal and perinatal morbidity. METHOD: A 38-year-old woman, Gravida 3 Para 1, with one previous miscarriage, presented with preterm labour at 33 weeks gestation. Antenatally, she was referred to a feto-maternal specialist for finding a placental tumour size 12 × 10 cm. Features are consistent with placental chorioangioma with polyhydramnios. The anomaly scan was normal. Antenatal fetal surveillance with Doppler studies were normal. RESULTS: During this admission, corticosteroid was given together with a tocolytic agent and opioid analgesia. Unfortunately, the labour progressed, and the patient felt reduced in fetal movement. The cardiotograph showed suspicious tracing. We proceed with emergency caesarean delivery. The placenta was sent for histopathology assessment which confirmed a large placental chorioangioma. The baby was born with Apgar's score of 9 at 1 min, pH of 7.28 and lactate of 7.28 with anaemia and thrombocytopenia. The uterus developed intermittent uterine atony, and the uterotonic agent was given. She recovered well post-delivery. The baby was admitted to the neonatal intensive care unit (NICU) and received a blood product transfusion and discharged from NICU on day 15 of life. DISCUSSION: Large placental chorioangioma is associated with polyhydramnios, preterm labour, postpartum haemorrhage, fetal anaemia, fetal distress, fetal hydrops and possible perinatal death. Multidisciplinary team involvement with feto-maternal specialists, anaesthetic and neonatologists would improve the outcome of both mother and fetus.

Intrauterine transfusion under fetal analgesia: the evaluation of perinatal outcomes.

Introduction: Intrauterine transfusion is the treatment for fetal anemia resulting from maternal alloimmunization, infections (parvovirus B19 and cytomegalovirus), single demise of a monochorionic twin, chorioangioma, and other rare conditions. Fetal analgesia is mandatory to reduce movement and pain perception during the procedure. This study aims to evaluate perinatal outcomes for such procedures, following the routine use of fetal analgesia in our clinical practice. Materials and methods: Retrospective analysis of cases from 2009 to 2022, including all confirmed fetal anemia with fetal blood sampling. After fetal analgesia, Rh-negative concentrated red blood cells were transfused, with ultrasonographic follow-up 24 h and 1 week later. In case of suspected brain lesion, magnetic resonance imaging was performed. Elective delivery was considered in case of persistent anemia after 34 weeks. Post-natal follow-up and comprehensive obstetric and perinatal outcomes data were collected. Results: Altogether 59 anemic fetuses were included, with 34 (57.6%) being hydropic. The causes of anemia were maternal alloimmunization (22, 37.3%), infections (13, 22%), monochorionicity (10, 16.9%), rare conditions (9, 15.3%), and two chorioangiomas (3.4%). The median gestational age at the procedure was 25.2 weeks (18-32 weeks), with no related preterm premature rupture of membranes (<48 h), or side effects from fetal analgesia. Gestational age at delivery was 33 weeks (26-41 weeks), with survival rate of 90%. There were four fetal demises, two termination of pregnancies, and eight neonatal deaths due to persistent severe anemia after preterm delivery. The main contributors to adverse outcome were the type of anemia, and the management with a preterm delivery. Conclusion: Intrauterine transfusion of red blood cells under analgesia is safe, with low incidence of obstetric complication.

Multiple giant placental chorioangioma: A case report.

Key Clinical Message: Giant chorioangiomas, despite being rare, pose significant fetal and maternal risks. Timely and individualized treatment plans are crucial to reduce morbidity and mortality when fetal compromise occurs. Additionally, successful conservative management relies on consistent ultrasound monitoring, Doppler flowmetry assessments, and amniotic fluid level measurements. Abstract: Chorioangiomas are benign placental tumors that manifest in approximately 1% of pregnancies. Giant chorioangiomas, characterized by tumors exceeding 4 cm, are exceptionally rare and pose substantial risks to maternal and fetal health. This case report details a patient with multiple giant chorioangiomas, emphasizing the rarity and consequential complications associated with these tumors. A 23-year-old woman, G3P2, at 28 weeks gestational age, was diagnosed with multiple large, well-defined placental masses with increased vascularity, indicative of giant placental chorioangiomas. Subsequent ultrasound revealed various fetal anomalies such as cleft palate and lip, as well as lung and heart abnormalities. At 34+5 weeks of gestation, an emergency cesarean section was performed due to preeclampsia. Subsequently, a female neonate was born with hydrops fetalis. Unfortunately, she passed away within the first hour of her life. Complications associated with chorioangiomas primarily arise from arteriovenous shunts, which potentially lead to compromised fetal perfusion and cardiac failure. Although small-sized chorioangiomas are often discovered incidentally, Doppler ultrasound and magnetic resonance imaging can reliably distinguish these tumors from other placental lesions. Additionally, management strategies tailored to gestational age and maternal-fetal symptoms typically necessitate a multidisciplinary approach. However, additional research is essential to understand the mechanisms of chorioangiomas and to develop comprehensive management guidelines.

A singleton pregnancy with placental chorioangioma and hydrops fetalis complicated with mirror syndrome and ritodrine-induced side effects: a case report.

BACKGROUND: Ritodrine hydrochloride is a widely used beta-adrenergic agonist used to stop preterm labor in Taiwan. Many side effects causing maternal morbidity and mortality have been reported. We report a case complicated with ritodrine-induced side effects and mirror syndrome that was associated with placental chorioangioma. CASE PRESENTATION: A 36-year-old singleton pregnant woman at 25 6/7 weeks of gestation, with an undiagnosed placental chorioangioma, underwent tocolysis due to preterm uterine contractions. Her clinical condition deteriorated, attributed to mirror syndrome and adverse events induced by ritodrine. An emergency cesarean section was performed at 27 1/7 weeks of gestation, delivering an infant with generalized subcutaneous edema. A placental tumor measuring 8.5 cm was discovered during the operation, and pathology confirmed chorioangioma. Gradual improvement in her symptoms and laboratory data was observed during the postpartum period. Identifying mirror syndrome and ritodrine-induced side effects poses challenges. Therefore, this case is educational and warrants discussion. CONCLUSION: Our case demonstrates mirror syndrome induced by chorioangioma, which is rare, and ritodrine-induced side effects. The cessation of intravenous ritodrine and delivery are the best methods to treat maternal critical status due to fluid overload.

Rare non-trophoblastic vascular placental tumor: A diagnostic challenge.

Key Clinical Message: Pathologists should consider the differential diagnoses of placental chorioangioma, which encompass chorangiosis, chorangiomatosis, chorangiocarcinoma, intervillous thrombus, and placental infarct. Abstract: Chorioangiomas are rare non-trophoblastic vascular neoplasms of the placenta, occurring in approximately 1% of pregnancies. Large chorioangiomas (>4 cm) are infrequent and associated with an increased risk of pregnancy complications. There are limited reports in the literature regarding uncomplicated cases of large placental chorioangiomas.

Giant Chorioangioma of the Placenta as a Cause of Maternal, Foetal, and Neonatal Complications.

Chorioangioma is a benign placental neoplasm seen in about one percent of all pregnancies. The larger neoplasms generally cause severe foeto-maternal complications. We are reporting a case of a 33-year-old gravida three para two female who was incidentally diagnosed with chorioangioma at her routine 28-week antenatal follow-up. She delivered a preterm small-for-gestational-age female baby at 34 weeks with complications. Therefore, an early diagnosis warrants a close follow-up and timely intervention for a better outcome of the pregnancy.

Management of large placental chorioangioma: two-port laser approach for fetal intervention.

OBJECTIVES: There is a paucity of literature providing evidence-based guidelines for the management of large placental chorioangioma (≥ 4 cm in diameter). The objectives of this study were to compare outcomes between patients managed expectantly and those undergoing in-utero intervention and to describe the different in-utero techniques used for cessation of blood flow to the tumor and the associated outcome. METHODS: This was a retrospective cohort study of 34 patients referred for the management of large placental chorioangioma in a single center between January 2011 and December 2022, who were managed expectantly or underwent in-utero intervention. In-utero intervention was performed when the fetus developed any signs of impending compromise, including high combined cardiac output (CCO), worsening polyhydramnios or abnormal fetal Doppler velocimetry findings. Interventions included radiofrequency ablation (RFA), interstitial laser ablation (ILA) and single-port or two-port fetoscopic laser photocoagulation (FLP). Treatment selection was dependent on the proximity of the tumor to the umbilical cord insertion (UCI) and placental location. The two-port technique was performed in patients with a chorioangioma with large feeding vessels (≥ 3 mm) located in the posterior placenta, in which one port was used for occlusion using bipolar forceps and the other port was used for laser photocoagulation of the feeding vessels downstream. The single-port technique was used for chorioangioma with small feeding vessels (< 3 mm) located in the posterior placenta. ILA or RFA was performed in cases with an anterior placenta. Supportive treatments, including amnioreduction and intrauterine transfusion (IUT), were performed for worsening polyhydramnios and suspected fetal anemia based on middle cerebral artery Doppler flow studies, respectively. Comparative statistical analysis between cases undergoing expectant management vs in-utero intervention was performed. Descriptive details were provided for patients who underwent in-utero intervention. RESULTS: Thirty-four cases of large chorioangioma were evaluated, of which 25 (73.5%) were managed expectantly and nine (26.5%) underwent intervention. The frequency of polyhydramnios was significantly higher in the intervention group compared with the expectant-management group (66.7% vs 8.0%, P < 0.001). The live-birth rate among expectantly managed cases with large chorioangioma was significantly higher compared with that in cases that underwent in-utero intervention (96.0% vs 62.5%, P = 0.01). In the intervention group, preoperative CCO was elevated in all cases with available information and preoperative hydrops was present in 33.3% (3/9) of cases. One patient experienced fetal demise following IUT prior to planned FLP. Among the remaining eight patients, four underwent two-port FLP, two underwent single-port FLP, one underwent ILA and one underwent both ILA and RFA. All three cases in which hydrops was present at the time of intervention resulted in fetal demise. CONCLUSIONS: In-utero interventions aimed at cessation of blood flow in the feeding vessels are a therapeutic option for the management of cases with large chorioangioma. The two-port percutaneous technique appears to improve the efficiency of FLP when a large chorioangioma with large feeding vessels is located in the posterior placenta. We propose that in-utero interventions for large chorioangioma should be initiated prior to the development of fetal hydrops. © 2023 International Society of Ultrasound in Obstetrics and Gynecology.

Large Chorioangioma in Triplets: An Uncommon Occurrence.

A 25-year-old primigravida presented at 26 weeks of gestation by dates, the first time for the routine antenatal checkup. No histories were suggestive of pregnancy-induced hypertension (PIH) and edema. On physical examination, pallor was present with microcytic hypochromic anemia. Raised beta-human chorionic gonadotropin (HCG) and alpha-fetoprotein (AFP) levels were present. Ultrasonography revealed triples with two thin echogenic intertwining membranes. Anomaly scan did not reveal any abnormality in fetuses. The placenta showed a large oval hypoechoic mass arising from its edge and bulge into the amniotic fluid. A central feeding vessel with a branching pattern and pulsatile color flow like that of the umbilical artery is noted on the color Doppler. She was spontaneously preterm delivered vaginally at 28 weeks of gestation. All three fetuses were stillborn. Histopathological diagnosis of angiomatous chorioangioma was confirmed. This case classically represents a grave complication of the large chorioangioma.

Watchful Expectant Management of a Giant Placental Chorioangioma With Favorable Maternal and Fetal Outcomes: A Case Report.

Placental chorioangioma is the most common benign tumor of the placenta. However, clinically evident chorioangiomas are less common with an incidence ranging between 1:3,500 and 1:9,000 births. In the majority of cases, it is small or microscopic and of no clinical significance. If it increases more than 5 cm in size, then it may be associated with serious maternal complications such as mirror syndrome, polyhydramnios, preeclampsia, antepartum hemorrhage, preterm labor and delivery, and postpartum hemorrhage, as well as fetal complications such as fetal anemia, fetal thrombocytopenia, fetal hydrops, intrauterine growth restriction, fetal demise, and neonatal death. Giant chorioangioma associated with polyhydramnios leads to high perinatal morbidity and mortality. Chorioangioma with complications before fetal viability needs interventions. Conservative management with close surveillance can be considered in selective cases. We report a case of giant placental chorioangioma complicated with severe polyhydramnios managed conservatively with favorable maternal and fetal outcomes.

Giant Placental Chorangioma and Severe Ductal Arch Constriction: A Case Report With a Favorable Outcome.

Giant chorangiomas are uncommon yet frequently associated with adverse pregnancy outcomes. A 37-year-old female was referred due to findings of a placental mass during a second-trimester ultrasound. A fetal survey at 26 weeks revealed a 69×97×75 mm heterogenous placental tumor with two prominent feeding vessels. Her prenatal course was complicated by worsening polyhydramnios requiring amnioreduction, gestational diabetes, and transient severe ductal arch (DA) constriction. Placental pathology confirmed the diagnosis of giant chorioangioma following delivery at 36 weeks. To our knowledge, this represents the first case of DA constriction in the setting of a giant chorangioma.

Placental chorioangioma and pregnancy outcome: a ten-year retrospective study in a tertiary referral centre.

BACKGROUND: Placental chorioangioma is a rare disorder in pregnancy. We retrospectively reviewed the perinatal complications and long-term outcomes in pregnancies with placental chorioangioma and evaluated the factors affecting disease prognosis. METHODS: We reviewed pregnant women who delivered at our hospital in the past decade and whose diagnosis of placental chorioangioma was confirmed by pathological diagnosis. Information on maternal demographics, prenatal sonographic findings and perinatal outcomes was obtained by reviewing the medical records. In the latter part of the study, follow-up of children was conducted by phone interview. RESULTS: In the 10 years from August 2008 to December 2018, 175 cases(0.17%) were identified as placental chorioangioma histologically and 44(0.04%) of them were large chorioangiomas. Nearly one-third of cases with large chorioangiomas were associated with severe maternal and fetal complications or required prenatal intervention. Although one-fifth of fetuses/newborns complicated with large chorioangiomas were lost perinatally, the long-term prognosis for surviving fetuses was generally good. Further statistical analysis revealed that tumor size and location affect prognosis. CONCLUSION: Placental chorioangioma may cause an unfavorable perinatal outcome. Regular ultrasound monitoring can provide the tumor characteristics which can be referred to for predicting the tendency of those complications and indicate when intervention may be necessary. It is not clear which factors lead to complications with fetal damage as the main manifestation or polyhydramnios as the main manifestation.

Placental chorioangioma separation in third trimester after fetoscopic laser therapy: Report of a rare case.

A primigravida received fetoscopic laser photocoagulation treatment at 25+1 weeks gestation as a chorioangioma enlarged to 61 × 46 × 52 mm. However, a cesarean section was performed due to the chorioangioma separated from the placenta at 32+2 weeks gestation. As the chorioangioma's blood supply were blocked, it was possible to provide expectant treatment.

Novel homozygous GLDC variant causing late-onset glycine encephalopathy: A case report and updated review of the literature.

Glycine encephalopathy (MIM #605899) is an autosomal recessive inborn error of metabolism caused by pathogenic variants in three genes GLDC, AMT, GCSH encoding glycine cleavage enzyme system. We report an 8-year-old boy with late-onset glycine encephalopathy who harbors a novel homozygous GLDC likely pathogenic variant c.707G > A p.(Arg236Gln). Polyhydramnios was noted at fetal ultrasound. He displayed global developmental delay, craniofacial dysmorphism, convulsions. Our report expands the phenotypic and genetic spectrum of late-onset nonketotic hyperglycinemia.

Chorioangioma: a single tertiary care center retrospective study.

OBJECTIVES: Chorioangioma represents a challenge due to the rarity of the condition, paucity of sufficient management guidelines, and controversies regarding the best invasive fetal therapy option; most of the scientific evidence for clinical treatment has been limited to case reports. The aim of this retrospective study was to review the natural antenatal history, maternal and fetal complications, and therapeutic modalities used in pregnancies complicated with placental chorioangioma at a single Center. METHODS: This retrospective study was conducted at King Faisal Specialist Hospital and Research Center (KFSH&RC) in Riyadh, Saudi Arabia. Our study population included all pregnancies with ultrasound features of chorioangioma, or histologically confirmed chorioangiomas, between January 2010 and December 2019. Data were collected from the patients' medical records, including the ultrasound reports and histopathology results. All subjects were kept anonymous; case numbers were used as identifiers. Data collected by the investigators were entered into Excel worksheets in an encrypted format. A MEDLINE database was used to retrieve 32 articles for literature review. RESULTS: Over a 10-year period between January 2010 and December 2019, 11 cases of chorioangioma were identified. Ultrasound remains the gold standard for diagnosis and follow-up of the pregnancy. Seven of the 11 cases were detected by ultrasound, allowing proper fetal surveillance and antenatal follow-up. Of the remaining six patients, one underwent radiofrequency ablation, two underwent intrauterine transfusion for fetal anemia due to placenta chorioangioma, one had vascular embolization with an adhesive material, and two were managed conservatively until term with ultrasound surveillance. CONCLUSIONS: Ultrasound remains the gold standard modality for prenatal diagnosis and follow-up of pregnancies with suspected chorioangiomas. Tumor size and vascularity play a significant role in the development of maternal-fetal complications and the success of fetal interventions. To determine the superior modality of fetal intervention mandates more data and research; nevertheless, Fetoscopic Laser Photocoagulation and embolization with adhesive material seem to be a lead choice, with reasonable fetal survival.

Radio frequency ablation for the intrauterine treatment of giant placental chorioangioma associated with fetal compromise: A case report.

Giant placental chorioangiomas associated with fetal hyperdynamic circulation complications are rare to see. Here, we summarized a case of giant placental chorioangioma associated with fetal anemia and heart failure treated by radiofrequency ablation (RFA) combined with cordocentesis and intrauterine transfusion. The sonographic appearance of the placental chorioangioma was atypical which was isoechoic with unclear boundary. RFA was performed successfully at 27 weeks of gestation, when the chorioangioma has increased to 17.0 × 10.6 × 12.3 cm3 . Unfortunately, intrauterine fetal demise was found on the first day after operation. After induction of labor, it was pathologically confirmed as placental chorioangioma.

Placental chorioangioma with an emphasis on rare giant placental chorioangioma and associated maternal and perinatal outcome: Clinicopathological study in a single centre.

Context: Giant placental chorioangiomas (GPC) are exceedingly rare and harbour potential to cause feto-maternal complications with resultant morbidity. Aims & Materials and Methods: A retrospective study using details from Department of Obstetrics & Gynaecology and Pathology is done to study the various clinical and pathological features of placental chorioangiomas with a special emphasis on the rare GPCs and associated complications. Results: Over a period of 16 years, 20 cases were diagnosed as chorioangioma in our institution. 60% of these occurred in primigravida (n=12) and 71% cases carried a female foetus. Only 25% cases were > 30 years. Maternal and foetal complications occurred in 85% and 50% cases. Pre-term labour was the common maternal complication and foetal death/stillbirth was the most common foetal complication. There were 15 cases of GPC, 73% occurred in primigravida (n=11) and 75% of cases carried a female foetus. There were no cases of maternal death or recurrence. Primigravidity was associated with maternal complication in contrast to multigravidity (P = 0.049). Mean age of mothers with maternal complications and those without maternal complications reached statistical significance (P = 0.001). Though histologically all the cases were similar, calcification and infarction were seen exclusively in GPC cases. Conclusion: GPCs are rare and our data adds evidence to use 4cm as an optimum cut-off in the definition. GPCs were associated with a high percentage of primigravidity, female foetus, and poorer outcome of pregnancy. Routine examination of placenta in unexplained foetal/perinatal demise must be stressed to detect microscopic evidence of chorioangioma.

Intrauterine transfusion of a hydropic fetus with anemia due to a giant chorioangioma: A case report.

Giant chorioangiomas are a potentially life-threatening condition that may require intrauterine therapy. We describe a case of a large chorioangioma (>4cm) diagnosed at 30 weeks of gestation causing severe fetal anemia and hydrops. An intrauterine blood transfusion was performed at 31 weeks with reversal of the anemia and hydrops. The neonate was born at 37 weeks showing respiratory distress syndrome that required neonatal intensive care unit admission but was discharged at 30 days of life. Further evaluation at two months of age showed no signs of abnormal neurodevelopment. When timely indicated, intrauterine transfusion of a hydropic fetus with anemia due to a giant chorioangioma is a potentially life-saving therapy that shows good neurodevelopment of the surviving fetus.