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":"1 1","pages":""},"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":"104 1","pages":""},"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}
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":"71 1","pages":""},"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}
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":"66 1","pages":""},"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}
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":"28 1","pages":"0"},"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":"23 1","pages":""},"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":"61 1","pages":""},"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":"53 1","pages":"0"},"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}
Laura M. Seske, M. Mastroianni, Keith T. Aziz, Laura W. Lewallen
Abstract Objectives Neonatal compartment syndrome (NCS) occurs when increased pressure within the fasciocutaneous compartment decreases capillary perfusion, causing irreversible tissue damage from ischemia. NCS is a rare condition that requires prompt diagnosis and treatment. Diagnosing NCS is highly dependent on the examination, which can be difficult in newborns. Prompt recognition provides the best chance for good outcomes. Case presentation We present a case of NCS diagnosed and treated based on physical examination findings. Fetal ultrasonography showed bladder distension, bilateral hydroceles, urethra dilation, and abdominal urinary ascites concerning for lower urinary tract obstruction and possible bladder rupture. At 1 h after birth, examination of the infant’s left upper extremity showed no spontaneous movement, the hand and forearm appeared dusky, and the hand had a large blister with desquamation. No pulse distal to the antecubital fossa was detected via Doppler ultrasonography. The infant was diagnosed with NCS and underwent urgent fasciotomy. The clinical appearance and perfusion of the left upper extremity gradually improved. At four months of age, the wounds were healed and the patient had full passive range of motion of the left upper extremity. Recovery of active motion is ongoing. Conclusions The presence of blistering and desquamation should provoke suspicion for NCS. Once NCS is diagnosed, prompt intervention is necessary to reduce the risk of poor functional outcomes. This case highlights the need for increased awareness of the risk developing compartment syndrome in utero as part of the rare sequalae in infants with congenital anomalies of kidney and urinary tract.
{"title":"An unexpected case of neonatal compartment syndrome associated with congenital anomalies of kidney and urinary tract","authors":"Laura M. Seske, M. Mastroianni, Keith T. Aziz, Laura W. Lewallen","doi":"10.1515/crpm-2022-0020","DOIUrl":"https://doi.org/10.1515/crpm-2022-0020","url":null,"abstract":"Abstract Objectives Neonatal compartment syndrome (NCS) occurs when increased pressure within the fasciocutaneous compartment decreases capillary perfusion, causing irreversible tissue damage from ischemia. NCS is a rare condition that requires prompt diagnosis and treatment. Diagnosing NCS is highly dependent on the examination, which can be difficult in newborns. Prompt recognition provides the best chance for good outcomes. Case presentation We present a case of NCS diagnosed and treated based on physical examination findings. Fetal ultrasonography showed bladder distension, bilateral hydroceles, urethra dilation, and abdominal urinary ascites concerning for lower urinary tract obstruction and possible bladder rupture. At 1 h after birth, examination of the infant’s left upper extremity showed no spontaneous movement, the hand and forearm appeared dusky, and the hand had a large blister with desquamation. No pulse distal to the antecubital fossa was detected via Doppler ultrasonography. The infant was diagnosed with NCS and underwent urgent fasciotomy. The clinical appearance and perfusion of the left upper extremity gradually improved. At four months of age, the wounds were healed and the patient had full passive range of motion of the left upper extremity. Recovery of active motion is ongoing. Conclusions The presence of blistering and desquamation should provoke suspicion for NCS. Once NCS is diagnosed, prompt intervention is necessary to reduce the risk of poor functional outcomes. This case highlights the need for increased awareness of the risk developing compartment syndrome in utero as part of the rare sequalae in infants with congenital anomalies of kidney and urinary tract.","PeriodicalId":9617,"journal":{"name":"Case Reports in Perinatal Medicine","volume":"1 1","pages":""},"PeriodicalIF":0.1,"publicationDate":"2023-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"78647297","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}
Z. Fasoulakis, M. Chatziioannou, Antonios Koutras, M. Theodora, Afroditi Pegkou, Andreas Pampanos, George Daskalakis, P. Antsaklis
Abstract Objectives Milroy syndrome is a rare hereditary disorder characterized by congenital lymphedema, caused by mutations in the vascular endothelial growth factor receptor 3 (VEGFR3) gene. Case presentation We present a case report of a first-described mutation of a male fetus diagnosed prenatally with Milroy syndrome through amniocentesis. The fetus had bilateral lower limb edema, and genetic testing confirmed the diagnosis of Milroy syndrome. The patient was closely monitored throughout the pregnancy, and after delivery, the infant was managed with appropriate therapies, including compression garments and manual lymphatic drainage. The parents were provided with appropriate counseling and support. Conclusions This case highlights the significance of early detection and appropriate management of Milroy syndrome, which can lead to improved outcomes for affected infants.
{"title":"Prenatal diagnosis and management of Milroy syndrome: a case report","authors":"Z. Fasoulakis, M. Chatziioannou, Antonios Koutras, M. Theodora, Afroditi Pegkou, Andreas Pampanos, George Daskalakis, P. Antsaklis","doi":"10.1515/crpm-2023-0013","DOIUrl":"https://doi.org/10.1515/crpm-2023-0013","url":null,"abstract":"Abstract Objectives Milroy syndrome is a rare hereditary disorder characterized by congenital lymphedema, caused by mutations in the vascular endothelial growth factor receptor 3 (VEGFR3) gene. Case presentation We present a case report of a first-described mutation of a male fetus diagnosed prenatally with Milroy syndrome through amniocentesis. The fetus had bilateral lower limb edema, and genetic testing confirmed the diagnosis of Milroy syndrome. The patient was closely monitored throughout the pregnancy, and after delivery, the infant was managed with appropriate therapies, including compression garments and manual lymphatic drainage. The parents were provided with appropriate counseling and support. Conclusions This case highlights the significance of early detection and appropriate management of Milroy syndrome, which can lead to improved outcomes for affected infants.","PeriodicalId":9617,"journal":{"name":"Case Reports in Perinatal Medicine","volume":"10 1","pages":""},"PeriodicalIF":0.1,"publicationDate":"2023-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"82990568","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}
京公网安备 11010802042870号
京ICP备2023020795号-1
扫码关注我们
求助内容：
应助结果提醒方式：