Mixed chorangioma and leiomyoma of the placenta, with a brief review of nontrophoblastic placental lesions.

Chorangioma is the most common type of primary non-trophoblastic tumor of the placenta, usually identified incidentally on ultrasound or at delivery. Leiomyomas within the placenta have been described, though they are rare and usually of maternal origin. We present an unusual case of a placental tumor with combined histopathologic and immunohistochemical features of both chorangioma and leiomyoma. A 39-year-old woman was found to have an echogenic placental mass at 33 weeks of gestation on ultrasound, that was thought to be a chorangioma. They followed up weekly, and performed a cesarean section at 39 weeks, due to concern for intrauterine growth restriction. No fetal or maternal complications occurred. Grossly, a 9-cm, red-brown mass with a broad-based stalk was identified on the fetal surface of the placenta near the periphery. Microscopically, the lesion was found to display characteristic features of chorangioma, with vascular proliferation, which stained positive for CD34 and CD31. SMA and caldesmon immunohistochemical staining was also positive, highlighting the proliferation of smooth muscle throughout the neoplasm. Literature review revealed a single additional case with similar characteristics.

Hepatic and Adrenocortical Choristomas in the Placenta.

Background Hepatic and adrenocortical choristomas are unusual findings in the placenta. This meta-analysis includes our own case report and 23 previously reported cases. We searched for patterns of associated placental, fetal and maternal aberrations in order to determine whether these choristomas are clinically relevant. Case report: In our case, abortion was induced due to fetal central nervous system and renal malformations. In the placenta a hepatic choristoma (<0.1 cm), thrombangiitis obliterans and a single umbilical artery were found. Results: In the literature, the majority of lesions were ≤1.0 cm (n = 21/24, 87.5%) and two hepatic choristomas manifested within chorangiomas. In a subfraction of cases, we found an association with twin/triple pregnancies (n = 6/24, 25%) and heterogeneous non-hepatic/non-adrenal malformations in fetuses (n = 4/24, 17%). Conclusion: Hepatic and adrenocortical choristomas are benign, could be based on focal epigenetic changes and might be related to chorangiomas but are not associated with a particular disease pattern or risk profile.

Large Hemoglobin Differences at Birth in Monochorionic Twins with a Placental Chorangioma and Delayed Cord Clamping.

We report a case of a monochorionic diamniotic twin with an uncomplicated pregnancy, but with an unexpected large intertwin hemoglobin (Hb) difference at birth. Twin 1 was delivered vaginally and had an uneventful neonatal course. The umbilical cord of Twin 1 was clamped approximately 5 min after birth. After the birth of Twin 1, Twin 2 developed severe bradycardia and showed limited cardiac output on ultrasound, for which an emergency cesarean section was performed. A full blood count revealed an Hb of 20.1 g/dL for Twin 1 and 10.2 g/dL for Twin 2 (intertwin difference 9.9 g/dL). Reticulocyte counts were similar, 40‰ and 38‰, respectively. Placental examination revealed 10 vascular anastomoses, including one arterio-arterial anastomosis with a diameter of 1.4 mm. Additionally, a large chorangioma was present on the placental surface of Twin 2. There was no color difference on the maternal side of the placenta. Based on the reticulocyte count ratio and the placental characteristics, twin anemia polycythemia sequence was ruled out as the cause of the large intertwin Hb difference. In this report, we discuss the various potential causes that could explain the large intertwin Hb difference including the role of delayed cord clamping in Twin 1, and the role of a large chorangioma, which may have attracted blood from the fetal circulation of Twin 2.

Successful Treatment of Placental Chorangiomas by Ultrasound-Guided Percutaneous in utero Microcoil Embolization.

Placental chorangiomas can cause a high-output fetal state and increase neonatal morbidity and mortality. There is a paucity of data published describing the optimal treatment of these cases, and methods for occlusion to date include placement of vascular clips, bipolar cautery, injection of alcohol or surgical glue, interstitial laser, and microcoil embolization. We report 2 cases of prenatally diagnosed chorangiomas that caused a high-output fetal state and were successfully treated with microcoil embolization. This case series describes our technique and supports microcoil embolization as a potentially safe and effective antenatal treatment option in symptomatic chorangiomas.

Genetic Analysis of Copy Number Variation in Large Chorangiomas.

INTRODUCTION: Chorangioma (CA) is the most common nontrophoblastic, vascular tumor-like lesion of the placenta with a reported incidence of 0.5% to 1% in all examined placentas. The underlying molecular mechanisms of CAs are still poorly elucidated, and a systematic investigation of the genetic background of CAs has not previously been done. MATERIALS AND METHODS: Tissue biopsies from 8 large (>40 mm) histologically confirmed CAs and 8 unaffected matched placenta controls, along with standard control DNA samples were analyzed for large genomic deletions and duplications using array comparative genomic hybridization (array-CGH) method. RESULTS: Array-CGH analysis revealed no rare or novel copy number variants in the CA samples compared with either standard control DNA or unaffected placenta DNA from the same individual. DISCUSSION: In this study, a systematic genetic investigation of 8 large CAs failed to demonstrate any large-scale pathogenic genetic changes. This lack of association might support a nongenetic, nontumorous origin of these lesions; however, additional genetic studies focusing on smaller genomic alterations are required to fully assess any possible genetic contribution.

Placental Pathology in Beckwith-Wiedemann Syndrome According to Genotype/Epigenotype Subgroups.

OBJECTIVES: To evaluate the frequency of placental pathological lesions in Beckwith-Wiedemann syndrome (BWS), an overgrowth disorder that exhibits etiologic molecular heterogeneity and variable phenotypic expression. MATERIALS AND METHODS: The study included 60 BWS patients with a proven molecular diagnosis and a placental pathological examination. Placentomegaly, placental mesenchymal dysplasia (PMD), chorangioma/chorangiomatosis, and extravillous trophoblastic (EVT) cytomegaly were evaluated and their frequencies in the different molecular subgroups were compared. Immunohistochemistry and fluorescent in situ hybridization (FISH) were performed on EVT cytomegaly. RESULTS: Placentomegaly was found in 70.9% of cases, PMD in 21.7%, chorangioma/chorangiomatosis in 23.3%, and EVT cytomegaly in 21.7%; there was no significant intergroup difference. EVT cytomegaly showed loss of p57 expression, increased Ki67 proliferating index, and polyploidy on FISH analysis. CONCLUSIONS: There was no genotype/epigenotype-phenotype correlation concerning placental lesions in BWS. Diffuse EVT cytomegaly with polyploidy may represent a placental finding suggestive of BWS.

[Placentary chorangio-leiomyoma]. / Corangioleiomioma placentario.

Chorangiomas are the most common non-trophoblastic tumors, generally appearing as incidental findings at the moment of delivery. The presence of leiomyomas inside placental parenchyma has been also described sporadically. In these cases, leiomyomas were primary maternal uterine neoplasms incorporated into the placenta during pregnancy. This case report presents a very unusual case of mesenchymal lesion, located in the fetal surface of the placenta, characterized by combined histopathologic and immunohistochemical features of chorangioma and leiomyoma. A single case reported with these characteristics was found in the international scientific literature, named as "chorangioleiomyoma". To our knowledge, this is the first case reported in our country.

Acardiac twin pregnancies part II: Fetal risk of chorangioma and sacrococcygeal teratoma predicted by pump/acardiac umbilical vein diameters.

BACKGROUND: We recently published pump/acardiac umbilical venous diameter (UVD) ratios, representing the pump twin's excess cardiac output fraction, of 27 acardiac twin pregnancies. There was a clear separation between the 17 pump twins that had life-threatening complications and the 10 that did not. The hypothesis of this study is that placental chorangioma and sacrococcygeal teratoma (SCT), tumors whose perfusion also causes high-output complications, have the same fetal outcome as pump twins when perfusion of the tumor requires the same excess cardiac output fraction. METHODS: We compared the three fetoplacental circulations. Fetuses with a placental chorangioma and acardiac twin pregnancies both have their feeding artery and draining vein located at the placental cord insertion. In contrast, SCT lacks a prescribed feeding artery and draining vein. We, therefore, had to modify our model to assume that the diameter of the hypothetical draining vein is related to the flow difference between inferior vena cava and superior vena cava. The latter flow has been estimated sonographically and is the same as the inferior vena cava flow in the absence of an SCT. Furthermore, a simple modification accounts for the different location of the tumor with respect to the placental cord insertion. RESULTS: We propose to apply the clinical pump/acardiac UVD ratios to pregnancies complicated by placental chorangiomas and the modified pump/acardiac UVD ratios for SCT. CONCLUSION: Risk prediction of these rare fetal tumors may be possible based on application of data on excess cardiac output fractions from pump/acardiac UVD ratios and will require future clinical validation. Birth Defects Research (Part A) 106:733-738, 2016. © 2016 Wiley Periodicals, Inc.

[Pseudosarcomatous chorangioma: A macroscopic and microscopic atypical tumor]. / Chorangiome pseudosarcomateux : une tumeur atypique sur le plan macroscopique et microscopique.

We report a case of an unusual chorangioma in a 26-year-old gravida 2, para 1 female. The clinical course was complicated by premature birth at 34 weeks' gestation. The baby presented with congenital cardiac and renal malformations. The tumor was 11 cm in size, separated from the main placental mass and exhibited atypical histologic characteristics such as fibromatous areas, high cellularity, nuclear atypia and high mitotic index. These histologic features must not be interpreted as malignancy.

Placental Chorangiocarcinoma a Specific Histological Pattern of Uncertain Incidence and Clinical Impact: Systematic Review of the Literature.

Chorangiocarcinoma is a very rare and misdiagnosed placental neoplasm. The unique morphologic features of the lesion distinguish it from other trophoblastic tumors and vascular abnormalities. We present a systematic review of the literature to provide clarity on chorangiocarcinoma entity and biology. A literature search was carried out in December 2022 using the keywords "Placental chorangiocarcinoma", "Chorangioma", "Placenta", and "Throphoblast proliferation". Articles published from 1988 to 2022 were obtained from Scopus, Google Scholar, and PUBMED. In our review, we examined maternal age, gestational age at the time of delivery, parity, type of pregnancy, placental weight, ultrasound features of the placenta, macroscopic examination and tumor size, microscopic examination, immunostaining, maternal beta-human chorionic gonadotropin, fetal and maternal outcome. Eight manuscripts were detected. They are all case reports. The macroscopic characteristics of the lesions were represented by the presence of a grey-yellow-white color well-demarcated round nodule. Microscopically, all the authors described typical aspects of malignancy as a high rate of mitosis, nuclear atypia and necrotic areas. In some cases, the presence of AE1/AE3 cytoplasmic positivity, p63 nuclear staining, and beta-human chorionic gonadotropin (BHCG) were reported. A good fetal outcome was reported in all cases of newborns with normal birth weight, except one with fetal growth restriction. Maternal outcome was good in all cases except one with maternal lung metastasis three months after delivery. The clinical course has probably underestimated the real incidence of the pathology. Only greater knowledge of its histology and its clinical course will allow us to evaluate the real prevalence of the disease.

When a Chorangioma Becomes a Burden in Fetal Survival: A Reported Case with an Updated Literature Review.

Chorangioma is a rare non-trophoblastic benign vascular neoplasm originating from the primitive chorionic mesenchyme. Usually asymptomatic, it affects approximately 1% of female fetuses. We present the case of a giant placental chorangioma (GPC) in a preterm male pregnancy coexisting with a maternal neuroendocrine carcinoma. A 30-week primigravida was admitted to the Obstetrics and Gynecology Clinic of the Targu-Mures Emergency Clinical Hospital, with abdominal discomfort, and an emergency C-section was performed for fetal congestive heart failure. Medical history revealed an advanced-stage rectal neuroendocrine carcinoma. At 20th gestational week, a well-vascularized placental mass was diagnosed. A 1500g premature male fetus was delivered. Histopathologically, the placental mass revealed an unencapsulated but well-circumscribed tumor with lobular architecture composed of congested vascular capillaries and thin-walled vessels. Diagnosis of giant placental chorangioma (GPC) was rendered. GPC is a challenging condition typically occurring in hypertensive or diabetic primigravidas with female fetuses. Antenatal management is suggested at an early stage for a desirable perinatal outcome.

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.

An unusual case of placental chorangioma and intrauterine death: Implications for proper antenatal care and prompt diagnosis.

We present an unusual case of placental chorangioma in a 32-year-old female with poor antenatal care. Abdominopelvic ultrasound revealed polyhydramnios with no fetal cardiac activity and suspected placental cyst. An emergency caesarean was performed, and she had still birth of male baby. The cut specimen of the placenta revealed a well-circumscribed marginal mass of 4 cm. Our case emphasizes the importance of regular antenatal screening for early detection of placental abnormalities. While chorangiomas are rare, they should be considered in the differential diagnosis of placental masses. Prompt diagnosis and appropriate management are essential to reduce the maternal and fetal complications associated with chorangiomas. Histological examination of the placenta plays a vital role in differentiating chorangioma from other placental abnormalities with different clinical implications.

Multiple Chorangioma Following Long-Term Secondary Infertility: A Rare Case Report and Review of Pathologic Differential Diagnosis.

Chorangioma (placental hemangioma) is a benign non-trophoblastic neoplasm of the placenta. Small chorangiomas are usually asymptomatic, but the giant and multiple ones rarely have a favorable outcome. We report a case of 29 weeks of gestational age (after long-term secondary infertility) with premature labor pain and undiagnosed multiple chorangioma leading to hydrops fetalis and neonatal death. Here we report the clinicopathological features of our case and chorangioma in general, along with comparison of different vascular lesions of placenta in terms of incidence, risk factors, complications, histologic origin, macroscopic and light and electron microscopic features.

A mystery mass on the placenta.

Chorioangioma or chorangioma is a benign placental tumour which occurs in 1% of the pregnancies. Large lesions of more than 4-5 cm in size, also known as giant chorangiomas, are rare with the incidence of 1:3500 and 1:9000 birth. Unlike small tumours, the giant chorangiomas are highly associated with pregnancy complications. We report a case of multiple large chorangiomas resulting in an extremely preterm delivery. A 24-year-old primigravida presented at 25 weeks of gestation for threatened preterm labour. Transabdominal ultrasound revealed an echogenic mass on the placenta measuring 8.7 × 4.4 cm. Following a successful tocolysis and administration of a course of antenatal corticosteroids, she was discharged home. At 27 weeks of gestation, she developed a second episode of preterm contractions. Besides, the foetus was found to be small for gestational age. In the second episode of preterm contractions, intravenous magnesium sulfate infusion was commenced for foetal neuroprotection. Tocolysis was commenced for severe prematurity. She went into spontaneous preterm labour. Placenta examination revealed multiple solid masses with fleshy and congested dark red surface. A histopathological examination of the placenta confirmed the diagnosis of chorangiomas. The baby was discharged in good condition at 5 months of age. Placental chorangiomas, notably when they are multiple and large in size, are associated with adverse pregnancy outcomes. Therefore, close antenatal surveillance is necessary to allow timely recognition and intervention of pregnancy complications. Our case portrays an unexpected favourable neonatal outcome associated with a giant chorangiomas.

Successful Management of Giant Placental Chorangioma by Microcoil Embolization.

Optimal prenatal management of giant placental chorangioma (also known as chorioangioma, angiomyxoma, fibroangiomyxoma, or fibroma) has yet to be determined. Interventions intended to devascularize the tumor such as interstitial laser, bipolar coagulation, fetoscopic laser photocoagulation, and chemical embolization have met mixed results. We report a minimally invasive, extra-amniotic approach, technically similar to cordocentesis, of microcoil embolization of the feeding vessel. These percutaneously placed microcoils initiate clot formation at the site of insertion and are unable to migrate through the tumor, thereby minimizing fetal harm by downstream embolic phenomena. Intervention at 26 and 22 weeks resulted in intraoperative fetal loss in the former and vaginal delivery at term of a healthy neonate in the latter. Preoperative, intraoperative, and placental findings are highlighted. The ease and safety of this procedure may alter the risk-benefit equation toward earlier intervention with potentially better clinical outcomes.

Corioangioma en paciente multípara. Caso clínico / Chorioangioma in multiparous aged. Clinical case

Resumen ANTECEDENTES: El corioangioma es un tumor vascular de origen placentario perteneciente al grupo de los hemangiomas vasculares. Es el tumor placentario primario no trofoblástico más frecuente de origen vascular. CASO CLÍNICO: Paciente de 37 años con antecedentes de: menarquia a los 12 años, inicio de la vida sexual activa a los 13 años, tres embarazos y dos partos. Del embarazo actual solo había tenido dos consultas prenatales. El ultrasonido reportó 35 semanas de embarazo con feto de 2250 g. La paciente tuvo dolor de origen obstétrico y trabajo de parto en fase latente. El parto fue eutócico, sin complicaciones. Se observó una tumoración placentaria. CONCLUSIONES: La valoración placentaria mediante ultrasonido-Doppler de control es importante para detectar tumoraciones y evitar que, cuando son pequeñas, pasen inadvertidas, como los corioangiomas.

Abstract BACKGROUND: Chorioangioma is a vascular tumor of placental origin belonging to the group of vascular hemangiomas. It is the most frequent non-trophoblastic primary placental tumor of vascular origin. CLINICAL CASE: 37-year-old patient with a history of: menarche at 12 years of age, beginning of active sexual life at 13 years of age, three pregnancies and two deliveries. She had only had two prenatal visits for the current pregnancy. The ultrasound reported 35 weeks of pregnancy with a 2250 g fetus. The patient had pain of obstetric origin and labor in the latent phase. The delivery was euthecological, without complications. A placental tumor was noted. CONCLUSIONS: Placental assessment by control ultrasound-Doppler is important to detect tumors and to avoid that, when they are small, they go unnoticed, such as chorangiomas.

Exploring the association between chorangioma and infantile haemangioma in singleton and multiple pregnancies: a case-control study in a Swedish tertiary centre.

OBJECTIVES: Placenta or placental chorangioma could be the origin site of infantile haemangioma since they share various histochemical and genetic characteristics with placental vascular tissue. The aim of the current study was to investigate the association between chorangiomas and infantile haemangiomas in singleton and multiple pregnancies. MATERIALS AND METHODS: An informative questionnaire enquiring about the presence or not of infantile haemangioma and including illustrative photos of haemangioma was sent to 469 (153 cases with chorangioma and 316 controls) mothers of 323 singleton (104 cases and 219 controls) and 146 multiple (49 cases and 97 controls) liveborn neonates registered in Sweden. Overall, 310 mothers (66.1%) from 216 singleton and 94 multiple pregnancies (96 cases and 214 controls) provided feedback and their consent to participate in the current case-control study. RESULTS: The incidence of infantile haemangioma showed no statistically significant differences between cases and controls (18.8% vs 18.2%) or between singleton and multiple pregnancies (18.9% vs 17.0%). The frequency of pre-eclampsia was significantly higher in cases with chorangioma compared with controls (41.7% vs 24.3%, OR=2.22, 95% CI 1.33 to 3.71, p=0.0022) and in singleton compared with multiple pregnancies (33.3% vs 21.3%, OR=1.85, 95% CI 1.04 to 3.26, p=0.034), whereas there were no significant differences in the incidence of infantile haemangioma in neonates of mothers with or without pre-eclampsia or in neonates of mothers with multiple compared with singleton pregnancies. CONCLUSION: There was no association between placental chorangiomas and infantile haemangiomas. Multiple pregnancies or pre-eclampsia were not significantly related to higher incidence of infantile haemangioma.

Association of chorangiomas to hypoxia-related placental changes in singleton and multiple pregnancy placentas.

INTRODUCTION: Chorangiomas (CAs) are the most frequent non-trophoblastic tumor-like-lesions of the placenta, and since they occur with an unusual frequency in pregnancies at high altitude, they are considered as a part of a spectrum of hypoxia-related vascular lesions of the placenta. The aim of our study is to describe the morphological features of the CAs and to show associations between CAs and other hypoxia related morphological changes in placentas of singleton and multiple pregnancies. MATERIALS AND METHODS: Placentas from singleton (121 vs 242) and multiple (49 vs 98) pregnancies, with and without CAs, respectively, were selected from a cohort of 15,742 placentas and enrolled into a case control study. RESULTS: Singleton placentas with CAs showed increased incidence of hypoxia-related placental changes including accelerated maturation of chorionic villi (OR = 2.40, p < 0.001), infarction (OR = 2.89, p < 0.001), decidual arteriopathy (OR = 3.24, p < 0.001), fetal thrombosis (OR = 4.05, p < 0.001) and hypercoiled umbilical cords (OR = 5.55, p < 0.001). The incidence of CAs in multiple placentas was higher in our studied cohort and a significant associated change was shown with fetal thrombosis (OR = 4.58, p = 0.017). There were no significant morphological changes between CAs in singleton compared to multiple pregnancies. DISCUSSION: In singleton placentas, CA is associated with several placental changes related to hypoxia, whereas in multiple pregnancies this relationship is not present. We speculate that CAs in multiple pregnancies might reflect an adaptive mechanism for relative hypoxia per se in these pregnancies. CONCLUSION: Our study provides evidence that CAs are associated with an increased rate of hypoxia related changes in singleton placentas.

Atypical Cellular Chorangioma: A Potential Diagnostic Pitfall With Worrisome Aspects but a Favorable Prognosis.

Chorangiomas are rather frequent neoplasms encountered on placental examination but in rare cases they present some worrisome histological features that could alarm the pathologist and be misinterpreted as a malignant neoplasm, even if their biological behavior is favorable. We describe an unusual chorangioma with high cellularity and abundant mitosis that, after careful examination and postpartum follow-up, showed benign clinical course for mother and child confirming previous reported cases. This type of tumor is known in the literature as atypical cellular chorangioma and its identification is important in order to exclude potentially dangerous overtreatment.