Jack Hamer, Leo Gurney, Shireen Meher, Robert Negrine, V. Hodgetts Morton, Tamas Marton, R. K. Morris
Chorioangiomas are the most frequently occurring type of benign tumour of the placenta. However, large chorioangiomas greater than 4 cm are rare and can be more frequently associated with serious complications such as: polyhydramnios, hydrops fetalis, fetal anaemia, intrauterine growth restriction, preterm birth, and an increased risk of perinatal mortality. Importantly timely prenatal diagnosis with close surveillance alongside potential intrauterine intervention can prove impactful on pregnancy outcome and fetal survival. We present a case of a 36-year-old female referred to our tertiary fetal medicine unit at 28 weeks’ gestation with a large chorioangioma measuring 9.4×8.8×5.5 cm and ultrasonographic evidence of severe fetal anaemia and fetal hydrops. The patient underwent an intrauterine transfusion and in utero surgical therapy with radiofrequency ablation (RFA). Immediately following the procedure, the fetus sustained a period of bradycardia, followed by asystole. Delivery was expedited via emergency caesarean section. Careful planning and rapid delivery after fetal intervention within the most appropriate surgical setting mitigated risks for the baby and resulted in a positive outcome. The baby was discharged from the neonatal unit on day 84 of life. Large placental chorioangiomas are a rare occurrence, however, when associated with fetal complications present a high incidence of adverse perinatal outcomes. In utero interventions require careful planning and surgical expertise to ensure improved fetal and neonatal outcomes. To the best of our knowledge this case is the first recorded instance of a successful postnatal outcome following RFA for a large placental chorioangioma, whereby the fetus was complicated by fetal hydrops.
{"title":"Radiofrequency thermal ablation of giant placental chorioangioma complicated with fetal hydrops: a case report and successful outcome","authors":"Jack Hamer, Leo Gurney, Shireen Meher, Robert Negrine, V. Hodgetts Morton, Tamas Marton, R. K. Morris","doi":"10.1515/crpm-2023-0028","DOIUrl":"https://doi.org/10.1515/crpm-2023-0028","url":null,"abstract":"\u0000 \u0000 \u0000 Chorioangiomas are the most frequently occurring type of benign tumour of the placenta. However, large chorioangiomas greater than 4 cm are rare and can be more frequently associated with serious complications such as: polyhydramnios, hydrops fetalis, fetal anaemia, intrauterine growth restriction, preterm birth, and an increased risk of perinatal mortality. Importantly timely prenatal diagnosis with close surveillance alongside potential intrauterine intervention can prove impactful on pregnancy outcome and fetal survival.\u0000 \u0000 \u0000 \u0000 We present a case of a 36-year-old female referred to our tertiary fetal medicine unit at 28 weeks’ gestation with a large chorioangioma measuring 9.4×8.8×5.5 cm and ultrasonographic evidence of severe fetal anaemia and fetal hydrops. The patient underwent an intrauterine transfusion and in utero surgical therapy with radiofrequency ablation (RFA). Immediately following the procedure, the fetus sustained a period of bradycardia, followed by asystole. Delivery was expedited via emergency caesarean section. Careful planning and rapid delivery after fetal intervention within the most appropriate surgical setting mitigated risks for the baby and resulted in a positive outcome. The baby was discharged from the neonatal unit on day 84 of life.\u0000 \u0000 \u0000 \u0000 Large placental chorioangiomas are a rare occurrence, however, when associated with fetal complications present a high incidence of adverse perinatal outcomes. In utero interventions require careful planning and surgical expertise to ensure improved fetal and neonatal outcomes. To the best of our knowledge this case is the first recorded instance of a successful postnatal outcome following RFA for a large placental chorioangioma, whereby the fetus was complicated by fetal hydrops.\u0000","PeriodicalId":9617,"journal":{"name":"Case Reports in Perinatal Medicine","volume":null,"pages":null},"PeriodicalIF":0.1,"publicationDate":"2024-03-20","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140224643","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Alma Mackert, Xezal Derin, P. Agha-Mir-Salim, W. Henrich
Abstract Objectives Rhinoliquorrhea is a condition where cerebrospinal fluid (CSF) leaks due to a liquor fistula formation of traumatic or non-traumatic origin. It can be associated with increased intracranial pressure often due to idiopathic intracranial hypertension (IIH), typically found in young and obese female patients. Case presentation A 27-year-old woman, 2 gravida, 1 para, presented with clear rhinorrhoea. After a beta-trace-protein test the diagnosis of CSF leakage was determined. The woman had had a traumatic car accident in 2018 but had never developed clear rhinorrhoea, especially not in her first pregnancy after the accident. Due to stable condition of the mother further diagnostics were postponed until after the birth. An elective caesarean section was performed in 40 + 0 weeks of gestations. The structural bone defect in the posterior wall of the sphenoid sinus was surgically repaired by defect coverage postpartum. Conclusions Nasal CSF leakage in pregnancy has previously been described in four other case reports with mostly traumatic etiology. Additionally, IIH is an important diagnosis to keep in mind. So far there are no guidelines or evidence-based recommendations regarding to optimal fistula treatment of pregnant women available. For therapy a prophylactic antibiotic therapy, surgical reconstruction with sealing and a wait-and-see strategy should be considered and discussed.
{"title":"Spontaneous cerebrospinal fluid rhinorrhoea during pregnancy-case report and review of literature","authors":"Alma Mackert, Xezal Derin, P. Agha-Mir-Salim, W. Henrich","doi":"10.1515/crpm-2023-0006","DOIUrl":"https://doi.org/10.1515/crpm-2023-0006","url":null,"abstract":"Abstract Objectives Rhinoliquorrhea is a condition where cerebrospinal fluid (CSF) leaks due to a liquor fistula formation of traumatic or non-traumatic origin. It can be associated with increased intracranial pressure often due to idiopathic intracranial hypertension (IIH), typically found in young and obese female patients. Case presentation A 27-year-old woman, 2 gravida, 1 para, presented with clear rhinorrhoea. After a beta-trace-protein test the diagnosis of CSF leakage was determined. The woman had had a traumatic car accident in 2018 but had never developed clear rhinorrhoea, especially not in her first pregnancy after the accident. Due to stable condition of the mother further diagnostics were postponed until after the birth. An elective caesarean section was performed in 40 + 0 weeks of gestations. The structural bone defect in the posterior wall of the sphenoid sinus was surgically repaired by defect coverage postpartum. Conclusions Nasal CSF leakage in pregnancy has previously been described in four other case reports with mostly traumatic etiology. Additionally, IIH is an important diagnosis to keep in mind. So far there are no guidelines or evidence-based recommendations regarding to optimal fistula treatment of pregnant women available. For therapy a prophylactic antibiotic therapy, surgical reconstruction with sealing and a wait-and-see strategy should be considered and discussed.","PeriodicalId":9617,"journal":{"name":"Case Reports in Perinatal Medicine","volume":null,"pages":null},"PeriodicalIF":0.1,"publicationDate":"2023-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"84574225","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
I. Mazhoud, Wissal Skhiri, C. Hafsa, Amel Maghrebi, A. Ksiaa, M. Maatouk, A. Ben Salem
Abstract Objectives Fetus-in-fetu is a rare congenital anomaly that occur secondary to abnormal embryogenesis in a diamniotic monochorionic pregnancy. Its diagnosis can be accurately made by imaging ultrasonography, radiography, computed tomography, or magnetic resonance imaging. Differential diagnosis is an important issue because FIF, teratoma and cystic meconium peritonitis are very different in terms of their respective disease courses. Case presentation This is an interesting rare case of a 22-year-old pregnancy woman, presented for a routine antenatal ultrasound. The diagnosis of a fetus-in-fetu was suspected, complete surgical excision of the lesion was performed and the diagnosis was histopathologically confirmed Conclusions We describe also the common characteristic of FIF as revealed by prenatal and postnatal US, postnatal MRI, and the operative findings.
{"title":"Fetus-in-fetu: mimicking teratoma on antenatal ultrasound","authors":"I. Mazhoud, Wissal Skhiri, C. Hafsa, Amel Maghrebi, A. Ksiaa, M. Maatouk, A. Ben Salem","doi":"10.1515/crpm-2022-0024","DOIUrl":"https://doi.org/10.1515/crpm-2022-0024","url":null,"abstract":"Abstract Objectives Fetus-in-fetu is a rare congenital anomaly that occur secondary to abnormal embryogenesis in a diamniotic monochorionic pregnancy. Its diagnosis can be accurately made by imaging ultrasonography, radiography, computed tomography, or magnetic resonance imaging. Differential diagnosis is an important issue because FIF, teratoma and cystic meconium peritonitis are very different in terms of their respective disease courses. Case presentation This is an interesting rare case of a 22-year-old pregnancy woman, presented for a routine antenatal ultrasound. The diagnosis of a fetus-in-fetu was suspected, complete surgical excision of the lesion was performed and the diagnosis was histopathologically confirmed Conclusions We describe also the common characteristic of FIF as revealed by prenatal and postnatal US, postnatal MRI, and the operative findings.","PeriodicalId":9617,"journal":{"name":"Case Reports in Perinatal Medicine","volume":null,"pages":null},"PeriodicalIF":0.1,"publicationDate":"2023-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"81660448","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Orzeł M. Maria, Pruszek K. Weronika, Borek-Dzięcioł Beata, Głuszczak-Idziakowska Ewa, Kociszewska-Najman Bożena
Abstract Objectives The incidence of congenital brain tumors is estimated at 1.1–3.6 per 100.000 live births, accounting for 0.5–2 % of all cancers in the pediatric population. Congenital gliomas account for 3.1–8.9 % of all congenital brain tumors and are cancers with a poor prognosis. The rate of stillbirth and death on the first day of life reaches 29 %; 38 % die within the first week, and 56 % die within the first two months. The average length of survival is two years. Case presentation In the 29th week of pregnancy, a female fetus was diagnosed with intracranial hemorrhage complicated by hydrocephalus. Postnatal brain MRI imaging showed a solid proliferative lesion of the left hemisphere with dilatation of the ventricular system. Brown cerebrospinal fluid was collected during the puncture of the left lateral ventricle to reduce hydrocephalus. No tumor cells were detected by cytology. Due to increasing hydrocephalus, the patient was qualified for Rickham reservoir implantation. On day 27th, a craniotomy was performed to determine the etiology of recurrent prenatal intraventricular bleeding. During surgery, the bleeding mass raised the suspicion of neoplasm—histopathological examination of the retrieved tissue diagnosed WHO stage IV malignant glioma. The patient died at 8 months of age. Conclusions Prenatal diagnosis of an abnormal structure in the fetal brain remains a diagnostic challenge in neonates. Glioblastoma is a rare neoplasm with a poor prognosis.
{"title":"Congenital glioblastoma – prenatal diagnosis becoming a diagnostic challenge after birth: a case report","authors":"Orzeł M. Maria, Pruszek K. Weronika, Borek-Dzięcioł Beata, Głuszczak-Idziakowska Ewa, Kociszewska-Najman Bożena","doi":"10.1515/crpm-2023-0008","DOIUrl":"https://doi.org/10.1515/crpm-2023-0008","url":null,"abstract":"Abstract Objectives The incidence of congenital brain tumors is estimated at 1.1–3.6 per 100.000 live births, accounting for 0.5–2 % of all cancers in the pediatric population. Congenital gliomas account for 3.1–8.9 % of all congenital brain tumors and are cancers with a poor prognosis. The rate of stillbirth and death on the first day of life reaches 29 %; 38 % die within the first week, and 56 % die within the first two months. The average length of survival is two years. Case presentation In the 29th week of pregnancy, a female fetus was diagnosed with intracranial hemorrhage complicated by hydrocephalus. Postnatal brain MRI imaging showed a solid proliferative lesion of the left hemisphere with dilatation of the ventricular system. Brown cerebrospinal fluid was collected during the puncture of the left lateral ventricle to reduce hydrocephalus. No tumor cells were detected by cytology. Due to increasing hydrocephalus, the patient was qualified for Rickham reservoir implantation. On day 27th, a craniotomy was performed to determine the etiology of recurrent prenatal intraventricular bleeding. During surgery, the bleeding mass raised the suspicion of neoplasm—histopathological examination of the retrieved tissue diagnosed WHO stage IV malignant glioma. The patient died at 8 months of age. Conclusions Prenatal diagnosis of an abnormal structure in the fetal brain remains a diagnostic challenge in neonates. Glioblastoma is a rare neoplasm with a poor prognosis.","PeriodicalId":9617,"journal":{"name":"Case Reports in Perinatal Medicine","volume":null,"pages":null},"PeriodicalIF":0.1,"publicationDate":"2023-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"87506517","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Isabella Dávila Neri, Adriana Patricia Farias Vela, Rafael L. Aragón Mendoza, Roberto Gallo Roa, Giovanni Carlo Russo Vizcaino
Abstract Objectives Fraser syndrome is a rare congenital malformation characterized by cryptophthalmos, syndactyly and urogenital tract malformations. The association with hydrometrocolpos is infrequent, with only a few cases reported in the literature. Case presentation A 19-year-old primigravida presenting at 35 weeks of gestation, with prenatal finding of hydrometrocolpos associated with hypotelorism and microphthalmia. Pre-term cesarean delivery was performed due to breech labor and perinatal death. The autopsy confirmed hydrometrocolpos secondary to vaginal atresia and imperforate hymen, associated with cryptophthalmos, syndactyly, nasal and pinna malformations, confirming the diagnosis of Fraser syndrome. Conclusions Fraser syndrome is usually a postnatal diagnosis. The association with genital abnormalities explains the finding of hydrometrocolpos, which could be considered a diagnostic criterion for this syndrome.
{"title":"Prenatal hydrometrocolpos as an unusual finding in Fraser syndrome. Case report","authors":"Isabella Dávila Neri, Adriana Patricia Farias Vela, Rafael L. Aragón Mendoza, Roberto Gallo Roa, Giovanni Carlo Russo Vizcaino","doi":"10.1515/crpm-2022-0038","DOIUrl":"https://doi.org/10.1515/crpm-2022-0038","url":null,"abstract":"Abstract Objectives Fraser syndrome is a rare congenital malformation characterized by cryptophthalmos, syndactyly and urogenital tract malformations. The association with hydrometrocolpos is infrequent, with only a few cases reported in the literature. Case presentation A 19-year-old primigravida presenting at 35 weeks of gestation, with prenatal finding of hydrometrocolpos associated with hypotelorism and microphthalmia. Pre-term cesarean delivery was performed due to breech labor and perinatal death. The autopsy confirmed hydrometrocolpos secondary to vaginal atresia and imperforate hymen, associated with cryptophthalmos, syndactyly, nasal and pinna malformations, confirming the diagnosis of Fraser syndrome. Conclusions Fraser syndrome is usually a postnatal diagnosis. The association with genital abnormalities explains the finding of hydrometrocolpos, which could be considered a diagnostic criterion for this syndrome.","PeriodicalId":9617,"journal":{"name":"Case Reports in Perinatal Medicine","volume":null,"pages":null},"PeriodicalIF":0.1,"publicationDate":"2023-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"85973245","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Abstract Objectives Hydrometrocolpos is a pelvic cystic mass representing the distension of the vagina and uterus due to obstructive congenital anomalies of the female causing accumulation of fluid secretions in the vagina and endometrial cavity. Prenatal diagnosis is uncommon and usually noticed during the adolescent period for failure to see menses with cyclic abdominal pain, abdominal mass, and local compressive symptoms. Late diagnosis after delivery of newborns with this condition results in poor outcomes from local compressive symptoms. Case presentation Here, we present a case diagnosed with congenital hydrometrocolpos at 39 weeks of gestation during routine third-trimester ultrasound scanning. The newborn was delivered vaginally and huge hydrometrocolpos secondary to imperforate hymen was diagnosed postnatally, and a hymenectomy was done and the newborn was discharged and improved from the hospital. Conclusions Although congenital hydrometrocolpos occurs rarely, it is also better to suspect prenatally in a female fetus with a cystic pelvic mass. Antenatal ultrasound diagnosis of this condition will help to make decisions early and to prevent further complications which might occur both intrauterine and after birth.
{"title":"Antenatal ultrasound diagnosis of huge fetal hydrometrocolpos secondary to imperforate hymen and successful postnatal treatment: a case report","authors":"Mequanint Melesse Bicha, Zelalem Ayichew Workneh","doi":"10.1515/crpm-2023-0019","DOIUrl":"https://doi.org/10.1515/crpm-2023-0019","url":null,"abstract":"Abstract Objectives Hydrometrocolpos is a pelvic cystic mass representing the distension of the vagina and uterus due to obstructive congenital anomalies of the female causing accumulation of fluid secretions in the vagina and endometrial cavity. Prenatal diagnosis is uncommon and usually noticed during the adolescent period for failure to see menses with cyclic abdominal pain, abdominal mass, and local compressive symptoms. Late diagnosis after delivery of newborns with this condition results in poor outcomes from local compressive symptoms. Case presentation Here, we present a case diagnosed with congenital hydrometrocolpos at 39 weeks of gestation during routine third-trimester ultrasound scanning. The newborn was delivered vaginally and huge hydrometrocolpos secondary to imperforate hymen was diagnosed postnatally, and a hymenectomy was done and the newborn was discharged and improved from the hospital. Conclusions Although congenital hydrometrocolpos occurs rarely, it is also better to suspect prenatally in a female fetus with a cystic pelvic mass. Antenatal ultrasound diagnosis of this condition will help to make decisions early and to prevent further complications which might occur both intrauterine and after birth.","PeriodicalId":9617,"journal":{"name":"Case Reports in Perinatal Medicine","volume":null,"pages":null},"PeriodicalIF":0.0,"publicationDate":"2023-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"135261276","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Abstract Objectives With increasing rates of cesarean delivery across the United States, a trial of labor after cesarean (TOLAC) is a reasonable alternative for qualified candidates. Although Müllerian anomalies are associated with a variety of adverse pregnancy outcomes, there is little existing data regarding TOLAC in these patients. We present a case of a patient with a didelphys uterus who achieved a successful vaginal birth after cesarean section (VBAC) in the setting of labor augmentation. Case presentation Our patient is a 32-year-old G4P1021 (Gravida 4 Para 1,021–1 term delivery, 0 preterm deliveries, 2 abortions, 1 living offspring) who presented at 8 weeks of gestation with a known history of a didelphys uterus. Her obstetrical history was significant for a prior low-transverse cesarean section at term. All four of her pregnancies were located in the right uterine horn. At 39 weeks 3 days of gestation she presented in early labor and requested a TOLAC. She received an epidural, a cervical ripening balloon was placed, and she was started on pitocin. She pushed to deliver a viable infant. The patient’s postpartum course was uncomplicated, and she was discharged home on postpartum day two. Conclusions Müllerian anomalies are associated with several poor pregnancy outcomes including increased rates of PPROM, preterm delivery, FGR, and malpresentation necessitating a cesarean section. Our patient required augmentation of her labor but was ultimately able to achieve a successful VBAC with a healthy neonate. She represents an understudied population of patients with uterine anomalies who not only can have favorable pregnancy outcomes but may even be able to safely achieve a VBAC.
{"title":"A successful vaginal birth after cesarean in a patient with uterine didelphys","authors":"Samantha Gobioff, M. Plakogiannis, A. Grünebaum","doi":"10.1515/crpm-2023-0005","DOIUrl":"https://doi.org/10.1515/crpm-2023-0005","url":null,"abstract":"Abstract Objectives With increasing rates of cesarean delivery across the United States, a trial of labor after cesarean (TOLAC) is a reasonable alternative for qualified candidates. Although Müllerian anomalies are associated with a variety of adverse pregnancy outcomes, there is little existing data regarding TOLAC in these patients. We present a case of a patient with a didelphys uterus who achieved a successful vaginal birth after cesarean section (VBAC) in the setting of labor augmentation. Case presentation Our patient is a 32-year-old G4P1021 (Gravida 4 Para 1,021–1 term delivery, 0 preterm deliveries, 2 abortions, 1 living offspring) who presented at 8 weeks of gestation with a known history of a didelphys uterus. Her obstetrical history was significant for a prior low-transverse cesarean section at term. All four of her pregnancies were located in the right uterine horn. At 39 weeks 3 days of gestation she presented in early labor and requested a TOLAC. She received an epidural, a cervical ripening balloon was placed, and she was started on pitocin. She pushed to deliver a viable infant. The patient’s postpartum course was uncomplicated, and she was discharged home on postpartum day two. Conclusions Müllerian anomalies are associated with several poor pregnancy outcomes including increased rates of PPROM, preterm delivery, FGR, and malpresentation necessitating a cesarean section. Our patient required augmentation of her labor but was ultimately able to achieve a successful VBAC with a healthy neonate. She represents an understudied population of patients with uterine anomalies who not only can have favorable pregnancy outcomes but may even be able to safely achieve a VBAC.","PeriodicalId":9617,"journal":{"name":"Case Reports in Perinatal Medicine","volume":null,"pages":null},"PeriodicalIF":0.1,"publicationDate":"2023-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"81237266","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Adrita Khawash, R. Kronfli, A. Arasu, R. Gandhi, K. Nicolaides, A. Greenough
Abstract Objectives Bilateral congenital diaphragmatic hernias (CDH) occur in one to two percent of CDH patients. There is a lower survival due to the greater likelihood of lung hypoplasia and associated anomalies. We report an infant with bilateral CDH and duodenal atresia who was successfully treated by fetoscopic endoluminal tracheal occlusion (FETO). Case presentation The fetus was diagnosed with CDH at 23 weeks of gestation. Her mother was referred to our tertiary centre as the observed to expected lung-to-head ratio (O/E LHR) at 26 weeks of gestation was only 17 %. The fetus was treated by FETO with an increase in the LHR. The mother had polyhydramnios and underwent amniotic fluid drainage at 26 and 31 weeks of gestation. She had preterm, premature rupture of the membranes at 31+3 weeks of gestation. The FETO balloon was punctured and the mother received corticosteroids. She underwent spontaneous labour at 35+6 weeks of gestation when the LHR was 55 %. At birth, the female infant was electively intubated and ventilated. After successful stabilisation, surgical intervention was undertaken on day six when the defects were identified as bilateral, type C posterolateral CDHs. Bilateral patch repair of the CDHs was undertaken using ‘domed’ Goretex patches. Type one duodenal atresia (DA) was identified and repaired with enterotomy and diamond duodenoduodenostomy. There was partial and then full abdominal closure on days 12 and 15 respectively. The infant is now four months of age and requires no respiratory support. Conclusions FETO can improve prognosis in infants with bilateral CDH.
{"title":"Fetoscopic endoluminal tracheal occlusion (FETO) and bilateral congenital diaphragmatic hernia","authors":"Adrita Khawash, R. Kronfli, A. Arasu, R. Gandhi, K. Nicolaides, A. Greenough","doi":"10.1515/crpm-2023-0010","DOIUrl":"https://doi.org/10.1515/crpm-2023-0010","url":null,"abstract":"Abstract Objectives Bilateral congenital diaphragmatic hernias (CDH) occur in one to two percent of CDH patients. There is a lower survival due to the greater likelihood of lung hypoplasia and associated anomalies. We report an infant with bilateral CDH and duodenal atresia who was successfully treated by fetoscopic endoluminal tracheal occlusion (FETO). Case presentation The fetus was diagnosed with CDH at 23 weeks of gestation. Her mother was referred to our tertiary centre as the observed to expected lung-to-head ratio (O/E LHR) at 26 weeks of gestation was only 17 %. The fetus was treated by FETO with an increase in the LHR. The mother had polyhydramnios and underwent amniotic fluid drainage at 26 and 31 weeks of gestation. She had preterm, premature rupture of the membranes at 31+3 weeks of gestation. The FETO balloon was punctured and the mother received corticosteroids. She underwent spontaneous labour at 35+6 weeks of gestation when the LHR was 55 %. At birth, the female infant was electively intubated and ventilated. After successful stabilisation, surgical intervention was undertaken on day six when the defects were identified as bilateral, type C posterolateral CDHs. Bilateral patch repair of the CDHs was undertaken using ‘domed’ Goretex patches. Type one duodenal atresia (DA) was identified and repaired with enterotomy and diamond duodenoduodenostomy. There was partial and then full abdominal closure on days 12 and 15 respectively. The infant is now four months of age and requires no respiratory support. Conclusions FETO can improve prognosis in infants with bilateral CDH.","PeriodicalId":9617,"journal":{"name":"Case Reports in Perinatal Medicine","volume":null,"pages":null},"PeriodicalIF":0.1,"publicationDate":"2023-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"74120136","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Taha F. Hassan, Ryan D. Morgan, Akshay Raghuram, Benedicto C. Baronia
Abstract Objectives This article outlines an unusual presentation of a premature infant born from a pre-eclamptic mother born with a presentation resembling a disseminated intravascular coagulation syndrome. Case presentation Pregnancy-induced hypertension, also known as pre-eclampsia, and premature birth pose significant risks to neonates, making the fetus more susceptible to immunodeficiencies and coagulopathies. This article highlights a premature infant born to a pre-eclamptic mother with multiple complications. Our case involved jaundice, neonatal meningitis, thrombocytopenia, leukopenia, neutropenia, hemorrhage, apnea, gastrointestinal defects, and periventricular leukomalacia. Often these complications are seen immediately after birth; these symptoms may present after a certain amount of time lapses if the neonates if afflicted with malignancy or a viral, fungal, or bacterial infection. Here we describe the case of a premature neonate born to a preeclamptic mother that experienced these complications one day after her birth. Conclusions This is the first known case of an infant experiencing a “DIC-like” syndrome without any diagnosis of a primary hematological malignancy or infection after a certain amount of time had lapsed since her birth. As complications in premature infants as well as those from pre-eclamptic mothers are common, this case report highlights a successful model of care. We also explore the effect of a peri-COVID setting on the presentation of this patient, as similar cases have occurred post-COVID-19.
{"title":"DIC-like syndrome in a post-pre-eclampsia birth in a premature infant in a peri-COVID scenario","authors":"Taha F. Hassan, Ryan D. Morgan, Akshay Raghuram, Benedicto C. Baronia","doi":"10.1515/crpm-2023-0016","DOIUrl":"https://doi.org/10.1515/crpm-2023-0016","url":null,"abstract":"Abstract Objectives This article outlines an unusual presentation of a premature infant born from a pre-eclamptic mother born with a presentation resembling a disseminated intravascular coagulation syndrome. Case presentation Pregnancy-induced hypertension, also known as pre-eclampsia, and premature birth pose significant risks to neonates, making the fetus more susceptible to immunodeficiencies and coagulopathies. This article highlights a premature infant born to a pre-eclamptic mother with multiple complications. Our case involved jaundice, neonatal meningitis, thrombocytopenia, leukopenia, neutropenia, hemorrhage, apnea, gastrointestinal defects, and periventricular leukomalacia. Often these complications are seen immediately after birth; these symptoms may present after a certain amount of time lapses if the neonates if afflicted with malignancy or a viral, fungal, or bacterial infection. Here we describe the case of a premature neonate born to a preeclamptic mother that experienced these complications one day after her birth. Conclusions This is the first known case of an infant experiencing a “DIC-like” syndrome without any diagnosis of a primary hematological malignancy or infection after a certain amount of time had lapsed since her birth. As complications in premature infants as well as those from pre-eclamptic mothers are common, this case report highlights a successful model of care. We also explore the effect of a peri-COVID setting on the presentation of this patient, as similar cases have occurred post-COVID-19.","PeriodicalId":9617,"journal":{"name":"Case Reports in Perinatal Medicine","volume":null,"pages":null},"PeriodicalIF":0.0,"publicationDate":"2023-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"135159550","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Celine Rohaert, A. Poleij, Chantal Quispel, M. de Jong, P. Ciet, Florian Cassel
Abstract Objectives Birth-related mechanical trauma to the newborn is an important issue and may be underestimated [Chaturvedi A, Chaturvedi A, Stanescu AL, Blickman JG, Meyers SP. Mechanical birth-related trauma to the neonate: an imaging perspective. Insights Imag 2018;9:103–18]. Risk factors for birth-related injuries include vacuum or forceps delivery, large size for gestational age and abnormal presentation before delivery [Gupta R, Cabacungan ET. Neonatal birth trauma: analysis of yearly trends, risk factors, and outcomes. J Pediatr 2021;238:174–80]. When a newborn has a soft tissue mass, there is a wide range of potential diagnoses, ranging from benign traumatic origins to aggressive phenotypes of malignant tumors [Thacker M. Benign soft tissue tumors in children. Orthop Clin N Am 2014;44:433–44]. Diagnosing a congenital tumor in a newborn creates uncertainty for parents and health care providers. Accurate imaging is crucial for distinguishing soft tissue mass origins. Case presentation A 32 weeks 6 days pregnant Caucasian woman was admitted after premature prelabor rupture of membranes (PPROM). Fetal ultrasound showed no abnormalities, the infant was born by a caesarean section. The delivery was complicated by the infant’s transverse position. A female infant was born with a large left-sided dorsal soft tissue mass at the thoracic level with elastic consistency, and multiple skin lacerations. A broad differential diagnosis was made. Additional imaging was suggestive for a posttraumatic swelling due to transverse position during birth. The mass decreased and disappeared over three days. Conclusions The diagnosis of a soft tissue mass in a newborn can be challenging. A birth-related trauma affecting the soft tissue should be considered, especially if prenatal ultrasound findings were normal. Malpresentation during birth is a significant risk factor. Accurate diagnostic imaging is important to do before conducting further diagnostic examinations. The time course of the mass, before and after birth, can aid in determining its origin.
【摘要】目的新生儿机械损伤是一个重要的问题，但可能被低估了[Chaturvedi A, Chaturvedi A, Stanescu AL, Blickman JG, Meyers SP.新生儿机械损伤:影像学视角]。[j].中国影像科学，2018;9:103-18。分娩相关损伤的危险因素包括真空或产钳分娩、胎龄过大和分娩前的异常表现[Gupta R, Cabacungan ET.新生儿出生创伤:年度趋势、危险因素和结局分析]。中华儿科杂志(英文版);2009;38(3):391 - 391。当新生儿有软组织肿块时，可能的诊断范围很广，从良性创伤起源到恶性肿瘤的侵袭性表型[Thacker M.儿童良性软组织肿瘤]。中华骨科杂志，2014;44(4):433 - 441。新生儿先天性肿瘤的诊断给父母和医疗保健提供者带来了不确定性。准确的影像是鉴别软组织肿块来源的关键。一例怀孕32周6天的白人妇女因早产胎膜破裂(PPROM)入院。胎儿超声检查未发现异常，婴儿通过剖腹产出生。婴儿的横卧位使分娩变得复杂。一女婴儿出生时在胸部水平有一个大的左侧背软组织肿块，弹性一致性，多处皮肤撕裂伤。作出了广泛的鉴别诊断。额外的影像学提示，创伤后肿胀由于横卧位在分娩。三天后，肿块减少并消失。结论新生儿软组织肿块的诊断具有挑战性。应考虑影响软组织的出生相关创伤，特别是如果产前超声检查结果正常。出生时表现不良是一个重要的危险因素。在进行进一步的诊断检查之前，准确的诊断成像是很重要的。胎儿出生前和出生后的时间过程有助于确定其起源。
{"title":"Birth-related soft tissue injury due to transverse malpresentation at delivery: a case report","authors":"Celine Rohaert, A. Poleij, Chantal Quispel, M. de Jong, P. Ciet, Florian Cassel","doi":"10.1515/crpm-2023-0009","DOIUrl":"https://doi.org/10.1515/crpm-2023-0009","url":null,"abstract":"Abstract Objectives Birth-related mechanical trauma to the newborn is an important issue and may be underestimated [Chaturvedi A, Chaturvedi A, Stanescu AL, Blickman JG, Meyers SP. Mechanical birth-related trauma to the neonate: an imaging perspective. Insights Imag 2018;9:103–18]. Risk factors for birth-related injuries include vacuum or forceps delivery, large size for gestational age and abnormal presentation before delivery [Gupta R, Cabacungan ET. Neonatal birth trauma: analysis of yearly trends, risk factors, and outcomes. J Pediatr 2021;238:174–80]. When a newborn has a soft tissue mass, there is a wide range of potential diagnoses, ranging from benign traumatic origins to aggressive phenotypes of malignant tumors [Thacker M. Benign soft tissue tumors in children. Orthop Clin N Am 2014;44:433–44]. Diagnosing a congenital tumor in a newborn creates uncertainty for parents and health care providers. Accurate imaging is crucial for distinguishing soft tissue mass origins. Case presentation A 32 weeks 6 days pregnant Caucasian woman was admitted after premature prelabor rupture of membranes (PPROM). Fetal ultrasound showed no abnormalities, the infant was born by a caesarean section. The delivery was complicated by the infant’s transverse position. A female infant was born with a large left-sided dorsal soft tissue mass at the thoracic level with elastic consistency, and multiple skin lacerations. A broad differential diagnosis was made. Additional imaging was suggestive for a posttraumatic swelling due to transverse position during birth. The mass decreased and disappeared over three days. Conclusions The diagnosis of a soft tissue mass in a newborn can be challenging. A birth-related trauma affecting the soft tissue should be considered, especially if prenatal ultrasound findings were normal. Malpresentation during birth is a significant risk factor. Accurate diagnostic imaging is important to do before conducting further diagnostic examinations. The time course of the mass, before and after birth, can aid in determining its origin.","PeriodicalId":9617,"journal":{"name":"Case Reports in Perinatal Medicine","volume":null,"pages":null},"PeriodicalIF":0.1,"publicationDate":"2023-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"90279654","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
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