Marina Ramos Pérez, Pedro Cabrera Vega, L. Urquía Martí, F. García-Muñoz Rodrigo
Abstract Objectives To describe an infrequent association of multiple injuries in relation to perinatal trauma and the challenge of clinical examination for proper diagnosis. Case presentation A male newborn was born at 41 weeks gestational age to a multipara diabetic mother, by forceps. Apgar 8/9. Upon admission, he exhibited axial hypotonia, right brachial paresis, a large left parietal cephalohematoma, but no skull fractures or orbital injuries. He also showed ocular misalignment with marked esotropia of the right eye, ptosis and mild anisocoria, suggesting right Horner syndrome, and left facial palsy. During the oculo-cephalic reflex examination he exhibited a normal adduction and impaired abduction of the right eye. The cranial MRI showed an extensive left parietal cephalohematoma, with internal foci of recent bleeding, and supra and infratentorial laminar subdural hematomas. No lesions were evidenced in the cavernous sinuses. The recovery of extra and intraocular motility at one month of life in our patient highlighted the transitory nature of the lesions and was reassuring for the parents. Conclusions This case highlights the importance of a detailed clinical examination in the initial evaluation of a newborn at neurological risk after birth trauma. The association of sixth cranial nerve palsy, Horner syndrome, facial palsy, and brachial plexus injury have been scarcely referred in this context and, if not adequately interpreted, might suggest erroneous diagnoses with very different prognoses.
{"title":"Transient congenital Horner syndrome and multiple peripheral nerve injury: a scarcely reported combination in birth trauma","authors":"Marina Ramos Pérez, Pedro Cabrera Vega, L. Urquía Martí, F. García-Muñoz Rodrigo","doi":"10.1515/crpm-2021-0025","DOIUrl":"https://doi.org/10.1515/crpm-2021-0025","url":null,"abstract":"Abstract Objectives To describe an infrequent association of multiple injuries in relation to perinatal trauma and the challenge of clinical examination for proper diagnosis. Case presentation A male newborn was born at 41 weeks gestational age to a multipara diabetic mother, by forceps. Apgar 8/9. Upon admission, he exhibited axial hypotonia, right brachial paresis, a large left parietal cephalohematoma, but no skull fractures or orbital injuries. He also showed ocular misalignment with marked esotropia of the right eye, ptosis and mild anisocoria, suggesting right Horner syndrome, and left facial palsy. During the oculo-cephalic reflex examination he exhibited a normal adduction and impaired abduction of the right eye. The cranial MRI showed an extensive left parietal cephalohematoma, with internal foci of recent bleeding, and supra and infratentorial laminar subdural hematomas. No lesions were evidenced in the cavernous sinuses. The recovery of extra and intraocular motility at one month of life in our patient highlighted the transitory nature of the lesions and was reassuring for the parents. Conclusions This case highlights the importance of a detailed clinical examination in the initial evaluation of a newborn at neurological risk after birth trauma. The association of sixth cranial nerve palsy, Horner syndrome, facial palsy, and brachial plexus injury have been scarcely referred in this context and, if not adequately interpreted, might suggest erroneous diagnoses with very different prognoses.","PeriodicalId":9617,"journal":{"name":"Case Reports in Perinatal Medicine","volume":null,"pages":null},"PeriodicalIF":0.1,"publicationDate":"2021-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"80892486","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}
D. A. Mazabanda López, D. Reyes Súarez, L. Urquía Martí, F. García-Muñoz Rodrigo
Abstract Objectives To describe an infrequent case of bacteremia by Rothia dentocariosa in a newborn and to discuss its potential pathogenicity. Case presentation R. dentocariosa is an aerobic or facultative anaerobic gram-positive bacillus, common in the human oral cavity that has been isolated in cases of endocarditis, pneumonia, endophthalmitis and peritonitis in adults. Infections in the fetus and newborn have been scarcely reported. We present a full-term newborn with prolonged rupture of membranes, acute elevation of C-reactive protein, and isolation of R. dentocariosa in the blood culture. Endocarditis was ruled out and the patient did well with five days of intravenous broad spectrum antibiotics. To date, only a previous case of sepsis by R. dentocariosa in a neonate with meconium aspiration syndrome and an antenatal death of a full-term fetus with hemorrhagic brain lesions probably related to R. dentocariosa have been reported. Conclusions Until more information about the pathogenicity of this germ in the newborn is available, an individualized approach and a close clinical control of the patient with infectious risk factors and bacteremia by R. dentocariosa seem to be prudent.
{"title":"Rothia dentocariosa bacteremia in the newborn: causative pathogen or contaminant?","authors":"D. A. Mazabanda López, D. Reyes Súarez, L. Urquía Martí, F. García-Muñoz Rodrigo","doi":"10.1515/crpm-2021-0026","DOIUrl":"https://doi.org/10.1515/crpm-2021-0026","url":null,"abstract":"Abstract Objectives To describe an infrequent case of bacteremia by Rothia dentocariosa in a newborn and to discuss its potential pathogenicity. Case presentation R. dentocariosa is an aerobic or facultative anaerobic gram-positive bacillus, common in the human oral cavity that has been isolated in cases of endocarditis, pneumonia, endophthalmitis and peritonitis in adults. Infections in the fetus and newborn have been scarcely reported. We present a full-term newborn with prolonged rupture of membranes, acute elevation of C-reactive protein, and isolation of R. dentocariosa in the blood culture. Endocarditis was ruled out and the patient did well with five days of intravenous broad spectrum antibiotics. To date, only a previous case of sepsis by R. dentocariosa in a neonate with meconium aspiration syndrome and an antenatal death of a full-term fetus with hemorrhagic brain lesions probably related to R. dentocariosa have been reported. Conclusions Until more information about the pathogenicity of this germ in the newborn is available, an individualized approach and a close clinical control of the patient with infectious risk factors and bacteremia by R. dentocariosa seem to be prudent.","PeriodicalId":9617,"journal":{"name":"Case Reports in Perinatal Medicine","volume":null,"pages":null},"PeriodicalIF":0.1,"publicationDate":"2021-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"80094500","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}
A. Fuchs, L. Harris, A. Huber, Mia Heiligenstein, C. Heiselman, K. Herrera, Diana Garretto
Abstract Objectives SARS-CoV-2 remains a pressing issue for our obstetric community during the current pandemic. We present a case of a 22-year-old nulliparous woman 31 weeks pregnant, with significant clinical SARS-CoV-2 disease, in the setting of negative nasopharyngeal PCR testing but positive IgG antibodies. Case presentation This was a 22-year-old patient 31 weeks pregnant who presented with fever, tachycardia, and subsequently preterm premature rupture of membranes (PPROM) and pulmonary emboli with multifocal pneumonia. The patient underwent three negative SARS-CoV-2 tests via nasopharyngeal PCR testing during her hospital stay as well as a negative workup for fever. After a cesarean section for worsening maternal status after 7 days of hospitalization, the patient was admitted for worsening clinical status to the SICU. At the time of SICU admission patient was found to have serum IgG positive antibodies and was managed with intubation, antibiotics, and anticoagulation. Patient eventually left hospital against medical advice on hospital day 16 on oral antibiotics but was found to be recovering well at later outpatient follow up. Conclusions Diagnosis of SARS-CoV-2 remains a complicated picture in the setting of testing limitations. This case highlights an antepartum clinical presentation of severe SARS-CoV-2 and recommends a high clinical suspicion for diagnosis of SARS-CoV-2 and initiation of treatment in the pregnant population, even in the presence of negative nasopharyngeal PCR testing.
{"title":"A case of severe SARS-CoV-2 infection with negative nasopharyngeal PCR in pregnancy","authors":"A. Fuchs, L. Harris, A. Huber, Mia Heiligenstein, C. Heiselman, K. Herrera, Diana Garretto","doi":"10.1515/crpm-2020-0075","DOIUrl":"https://doi.org/10.1515/crpm-2020-0075","url":null,"abstract":"Abstract Objectives SARS-CoV-2 remains a pressing issue for our obstetric community during the current pandemic. We present a case of a 22-year-old nulliparous woman 31 weeks pregnant, with significant clinical SARS-CoV-2 disease, in the setting of negative nasopharyngeal PCR testing but positive IgG antibodies. Case presentation This was a 22-year-old patient 31 weeks pregnant who presented with fever, tachycardia, and subsequently preterm premature rupture of membranes (PPROM) and pulmonary emboli with multifocal pneumonia. The patient underwent three negative SARS-CoV-2 tests via nasopharyngeal PCR testing during her hospital stay as well as a negative workup for fever. After a cesarean section for worsening maternal status after 7 days of hospitalization, the patient was admitted for worsening clinical status to the SICU. At the time of SICU admission patient was found to have serum IgG positive antibodies and was managed with intubation, antibiotics, and anticoagulation. Patient eventually left hospital against medical advice on hospital day 16 on oral antibiotics but was found to be recovering well at later outpatient follow up. Conclusions Diagnosis of SARS-CoV-2 remains a complicated picture in the setting of testing limitations. This case highlights an antepartum clinical presentation of severe SARS-CoV-2 and recommends a high clinical suspicion for diagnosis of SARS-CoV-2 and initiation of treatment in the pregnant population, even in the presence of negative nasopharyngeal PCR testing.","PeriodicalId":9617,"journal":{"name":"Case Reports in Perinatal Medicine","volume":null,"pages":null},"PeriodicalIF":0.1,"publicationDate":"2021-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"81894057","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}
Erick J. Bordón Sardiña, Cristina Romero Álvarez, Rocío Díaz de Bethencourt Pardo, L. Urquía Martí, F. García-Muñoz Rodrigo
Abstract Objectives To describe the association between the rapid normalization of pCO2 after intubation in a patient with severe bronchopulmonary dysplasia managed with permissive hypercapnia, with the risk of developing hypocalcemia and seizures, and to make health care providers aware of this risk in similar cases. Case presentation An extreme premature infant, born at 25 weeks of gestational age (GA), developed a severe bronchopulmonary dysplasia (BPD) and, after several extubation failures could be managed with non-invasive ventilation and permissive hypercapnia, with capillary pCO2 of up to 80 mmHg and pH >7.20. At 46 postmenstrual age (PMA) he was intubated because of severe hypercapnia and compensating metabolic alkalosis. About 20 h after intubation, after normalization of pH and pCO2, he developed hypocalcemia and seizures, that remitted after iCa normalization. A comparison between arterial and capillary blood gases showed a significantly greater correlation between pH and iCa in arterial than in capillary samples. Conclusions Our findings emphasize the importance of avoiding the abrupt reduction of pCO2 and the close monitoring of acute metabolic changes after its correction in chronic patients with permissive hypercapnia, as well as the potential superiority of arterial samples over capillaries to improve the precision of this control.
{"title":"Severe hypocalcemia and seizures after normalization of pCO2 in a patient with severe bronchopulmonary dysplasia and permissive hypercapnia","authors":"Erick J. Bordón Sardiña, Cristina Romero Álvarez, Rocío Díaz de Bethencourt Pardo, L. Urquía Martí, F. García-Muñoz Rodrigo","doi":"10.1515/crpm-2021-0039","DOIUrl":"https://doi.org/10.1515/crpm-2021-0039","url":null,"abstract":"Abstract Objectives To describe the association between the rapid normalization of pCO2 after intubation in a patient with severe bronchopulmonary dysplasia managed with permissive hypercapnia, with the risk of developing hypocalcemia and seizures, and to make health care providers aware of this risk in similar cases. Case presentation An extreme premature infant, born at 25 weeks of gestational age (GA), developed a severe bronchopulmonary dysplasia (BPD) and, after several extubation failures could be managed with non-invasive ventilation and permissive hypercapnia, with capillary pCO2 of up to 80 mmHg and pH >7.20. At 46 postmenstrual age (PMA) he was intubated because of severe hypercapnia and compensating metabolic alkalosis. About 20 h after intubation, after normalization of pH and pCO2, he developed hypocalcemia and seizures, that remitted after iCa normalization. A comparison between arterial and capillary blood gases showed a significantly greater correlation between pH and iCa in arterial than in capillary samples. Conclusions Our findings emphasize the importance of avoiding the abrupt reduction of pCO2 and the close monitoring of acute metabolic changes after its correction in chronic patients with permissive hypercapnia, as well as the potential superiority of arterial samples over capillaries to improve the precision of this control.","PeriodicalId":9617,"journal":{"name":"Case Reports in Perinatal Medicine","volume":null,"pages":null},"PeriodicalIF":0.1,"publicationDate":"2021-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"89338818","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}
D. Karçaaltıncaba, M. Ozdogan, E. Turgut, H. Ozdemir, G. Taçoy, G. Inan
Abstract Objectives The population of female heart transplant recipients of reproductive age is increasing and pregnancy follow-up of these patients is important. Case presentation A 30-year-old patient who had a heart transplant due to viral myocarditis became pregnant spontaneously. A close follow-up by a multidisciplinary team allowed a normal pregnancy without maternal or fetal complications and the delivery of a healthy infant. Conclusions Successful pregnancy outcomes are possible in women who had a heart transplant. Careful and close surveillance by a multidisciplinary team is mandatory.
{"title":"Successful pregnancy outcome in patient with cardiac transplantation","authors":"D. Karçaaltıncaba, M. Ozdogan, E. Turgut, H. Ozdemir, G. Taçoy, G. Inan","doi":"10.1515/crpm-2021-0041","DOIUrl":"https://doi.org/10.1515/crpm-2021-0041","url":null,"abstract":"Abstract Objectives The population of female heart transplant recipients of reproductive age is increasing and pregnancy follow-up of these patients is important. Case presentation A 30-year-old patient who had a heart transplant due to viral myocarditis became pregnant spontaneously. A close follow-up by a multidisciplinary team allowed a normal pregnancy without maternal or fetal complications and the delivery of a healthy infant. Conclusions Successful pregnancy outcomes are possible in women who had a heart transplant. Careful and close surveillance by a multidisciplinary team is mandatory.","PeriodicalId":9617,"journal":{"name":"Case Reports in Perinatal Medicine","volume":null,"pages":null},"PeriodicalIF":0.1,"publicationDate":"2021-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"80302928","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}
Marcela Toro-Bejarano, R. Mora, I. Timor-Tritsch, J. Vernon, A. Monteagudo, F. D’Antonio, K. Duncan
Abstract Objectives Uterine myomas are a frequent finding in reproductive age women with an estimated incidence 12–25%. 1. Treatment of uterine myomas to facilitate good pregnancy rates and outcome, such as hysteroscopic, laparoscopic, abdominal resection uterine artery embolization among others were evaluated in terms of pregnancy outcome. While the literature is replete of the pregnancy complication of uterine rupture after myomectomies, 2–4 there are very few publications evaluate a relatively rare pregnancy complication associated with placental implantation within the uterine cavity at the site of the previous myomectomy, namely the myomectomy scar pregnancy (MSP). Despite their relative rarity, this type of pathologically adherent placenta rightfully belongs to the well-known entity of placenta accreta spectrum (PAS). Case presentation We present a complicated case of MSP and review the available literature to raise attention to its clinical appearance, its prenatal diagnosis so appropriate intrapartum management can be planned. Conclusions Despite the rarity of MSP, continuous attention should be given at every single routinely scheduled or indication driven obstetrical US scan following myomectomies to evaluate the placental site implantation regardless of the route and technique of their initial surgical procedure.
{"title":"Myomectomy scar pregnancy ‒ a serious, but scarcely reported entity: literature review and an instructive case","authors":"Marcela Toro-Bejarano, R. Mora, I. Timor-Tritsch, J. Vernon, A. Monteagudo, F. D’Antonio, K. Duncan","doi":"10.1515/crpm-2021-0071","DOIUrl":"https://doi.org/10.1515/crpm-2021-0071","url":null,"abstract":"Abstract Objectives Uterine myomas are a frequent finding in reproductive age women with an estimated incidence 12–25%. 1. Treatment of uterine myomas to facilitate good pregnancy rates and outcome, such as hysteroscopic, laparoscopic, abdominal resection uterine artery embolization among others were evaluated in terms of pregnancy outcome. While the literature is replete of the pregnancy complication of uterine rupture after myomectomies, 2–4 there are very few publications evaluate a relatively rare pregnancy complication associated with placental implantation within the uterine cavity at the site of the previous myomectomy, namely the myomectomy scar pregnancy (MSP). Despite their relative rarity, this type of pathologically adherent placenta rightfully belongs to the well-known entity of placenta accreta spectrum (PAS). Case presentation We present a complicated case of MSP and review the available literature to raise attention to its clinical appearance, its prenatal diagnosis so appropriate intrapartum management can be planned. Conclusions Despite the rarity of MSP, continuous attention should be given at every single routinely scheduled or indication driven obstetrical US scan following myomectomies to evaluate the placental site implantation regardless of the route and technique of their initial surgical procedure.","PeriodicalId":9617,"journal":{"name":"Case Reports in Perinatal Medicine","volume":null,"pages":null},"PeriodicalIF":0.1,"publicationDate":"2021-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"80120330","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}
C. Lesmes Heredia, M. Comas Rovira, Anna Moreno Baró, Sílvia Pina Pérez, Marc Zamora Lapiedra, Manuel Corona Martínez
Abstract Objectives Doppler measurement of fetal middle cerebral artery peak systolic velocity (MCA-PSV) is used to screen fetuses at risk of developing fetal anemia. This screening tool has demonstrated to be accurate to estimate the likelihood of fetal anemia and it has been used in the management of fetuses at this risk. It is rare to find in the literature cases of increased MCA-PSV in the absence of fetal anemia. We present the case of an elevated MCA-PSV in the absence of common causes of fetal anemia. Case presentation A 30-year-old woman at 21 weeks of gestation was referred to our unit with a left fetal renal agenesis and single umbilical artery diagnosis. The patient had a poorly controlled type 1 Diabetes with a booking HbA1C of 9.1%. At 31 weeks of gestation, MCA-PSV was raised (>1.5 multiples of the median) in the absence of any cause of fetal anemia and discrepancy of ventricular size with normal fetal heart function were seen. In a subsequent scan, thick interventricular septum was seen and hypertrophic cardiomyopathy (HC) was suggested. Follow-up ultrasonography showed a persistent MCA-PSV in a large for gestation age (LGA) fetus with HC. Due to the prenatal diagnosis of persistent elevated MCA-PSV in LGA fetus with diagnoses of HC in a poorly controlled type 1 diabetic patient labor was induced after fetal lung maturation. Biventricular hypertrophic cardiomyopathy was detected after birth. Conclusions HC may be an explanation for increased MCA-PSV without fetal anemia.
{"title":"Elevated fetal middle cerebral artery peak systolic velocity in diabetes type 1 patient: a case report","authors":"C. Lesmes Heredia, M. Comas Rovira, Anna Moreno Baró, Sílvia Pina Pérez, Marc Zamora Lapiedra, Manuel Corona Martínez","doi":"10.1515/crpm-2021-0003","DOIUrl":"https://doi.org/10.1515/crpm-2021-0003","url":null,"abstract":"Abstract Objectives Doppler measurement of fetal middle cerebral artery peak systolic velocity (MCA-PSV) is used to screen fetuses at risk of developing fetal anemia. This screening tool has demonstrated to be accurate to estimate the likelihood of fetal anemia and it has been used in the management of fetuses at this risk. It is rare to find in the literature cases of increased MCA-PSV in the absence of fetal anemia. We present the case of an elevated MCA-PSV in the absence of common causes of fetal anemia. Case presentation A 30-year-old woman at 21 weeks of gestation was referred to our unit with a left fetal renal agenesis and single umbilical artery diagnosis. The patient had a poorly controlled type 1 Diabetes with a booking HbA1C of 9.1%. At 31 weeks of gestation, MCA-PSV was raised (>1.5 multiples of the median) in the absence of any cause of fetal anemia and discrepancy of ventricular size with normal fetal heart function were seen. In a subsequent scan, thick interventricular septum was seen and hypertrophic cardiomyopathy (HC) was suggested. Follow-up ultrasonography showed a persistent MCA-PSV in a large for gestation age (LGA) fetus with HC. Due to the prenatal diagnosis of persistent elevated MCA-PSV in LGA fetus with diagnoses of HC in a poorly controlled type 1 diabetic patient labor was induced after fetal lung maturation. Biventricular hypertrophic cardiomyopathy was detected after birth. Conclusions HC may be an explanation for increased MCA-PSV without fetal anemia.","PeriodicalId":9617,"journal":{"name":"Case Reports in Perinatal Medicine","volume":null,"pages":null},"PeriodicalIF":0.1,"publicationDate":"2021-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"79541059","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}
L. Sá, T. Pereira, Ana B. Ferreira, A. Reis, A. Madureira, Margarida Azevedo
Abstract Objectives The aim of this article is to describe a clinical case of Scimitar syndrome, an unusual cause of respiratory distress in the neonatal period. This syndrome is a rare form of partial or total anomalous right lung venous drainage to the inferior caval vein, usually associated with right pulmonary hypoplasia. Case presentation The authors report a case of a 36-week-old newborn admitted to the neonatal intensive care unit with worsening respiratory distress. Chest radiography revealed a characteristic right pulmonary hypoplasia and a tubular structure paralleling the right heart border in the shape of a Turkish sword (“scimitar”). Chest computed tomography angiography confirmed the diagnosis. Conclusions Age of presentation and the severe malformations found, contributed to the patient’s poor outcome in this case.
{"title":"Scimitar syndrome – a case report","authors":"L. Sá, T. Pereira, Ana B. Ferreira, A. Reis, A. Madureira, Margarida Azevedo","doi":"10.1515/crpm-2020-0088","DOIUrl":"https://doi.org/10.1515/crpm-2020-0088","url":null,"abstract":"Abstract Objectives The aim of this article is to describe a clinical case of Scimitar syndrome, an unusual cause of respiratory distress in the neonatal period. This syndrome is a rare form of partial or total anomalous right lung venous drainage to the inferior caval vein, usually associated with right pulmonary hypoplasia. Case presentation The authors report a case of a 36-week-old newborn admitted to the neonatal intensive care unit with worsening respiratory distress. Chest radiography revealed a characteristic right pulmonary hypoplasia and a tubular structure paralleling the right heart border in the shape of a Turkish sword (“scimitar”). Chest computed tomography angiography confirmed the diagnosis. Conclusions Age of presentation and the severe malformations found, contributed to the patient’s poor outcome in this case.","PeriodicalId":9617,"journal":{"name":"Case Reports in Perinatal Medicine","volume":null,"pages":null},"PeriodicalIF":0.1,"publicationDate":"2021-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"76859910","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}
Kaitlyn M. Lopushinsky, A. Harabor, Jaya P. Bodani
Abstract Objectives To describe the case of a preterm infant with intrauterine HSV infection. To discuss the epidemiology, features and complications associated with intrauterine HSV infection by conducting a review of medical literature. Case presentation A preterm male infant was born with diffuse skin erosions in various stages of healing suggestive of denuded bullae. Prenatal ultrasound showed evolving fetal cerebral ventriculomegaly, suspected Dandy-Walker malformation, and oligohydramnios. Neonatal skin swabs were positive for herpes simplex virus type 2 by polymerase chain reaction and the patient was treated with intravenous acyclovir from birth. Cranial ultrasound and magnetic resonance imaging findings showed severe progressive ex-vacuo ventricular dilatation consistent with intrauterine herpes simplex infection. Due to those findings and progressive multi-organ dysfunction, care was redirected to a palliative path and the child expired at 21 days of age. In retrospect, the mother had a few atypical vesicles on the left hand 4–5 weeks prior to delivery with a presumptive diagnosis of pompholyx. Conclusions Although confirmed intrauterine herpes simplex virus (HSV) infections are very rare, the potentially devastating prognosis and complications as evidenced by our case show that awareness of intrauterine HSV is critical for pediatricians as well as physicians involved in antenatal care. In order to adequately assess, treat and counsel pregnant women, more work is required to detect HSV infection, evaluate possible congenital infection and improve outcomes with fetal and neonatal treatment.
{"title":"No footprint too small: case of intrauterine herpes simplex virus infection","authors":"Kaitlyn M. Lopushinsky, A. Harabor, Jaya P. Bodani","doi":"10.1515/crpm-2021-0047","DOIUrl":"https://doi.org/10.1515/crpm-2021-0047","url":null,"abstract":"Abstract Objectives To describe the case of a preterm infant with intrauterine HSV infection. To discuss the epidemiology, features and complications associated with intrauterine HSV infection by conducting a review of medical literature. Case presentation A preterm male infant was born with diffuse skin erosions in various stages of healing suggestive of denuded bullae. Prenatal ultrasound showed evolving fetal cerebral ventriculomegaly, suspected Dandy-Walker malformation, and oligohydramnios. Neonatal skin swabs were positive for herpes simplex virus type 2 by polymerase chain reaction and the patient was treated with intravenous acyclovir from birth. Cranial ultrasound and magnetic resonance imaging findings showed severe progressive ex-vacuo ventricular dilatation consistent with intrauterine herpes simplex infection. Due to those findings and progressive multi-organ dysfunction, care was redirected to a palliative path and the child expired at 21 days of age. In retrospect, the mother had a few atypical vesicles on the left hand 4–5 weeks prior to delivery with a presumptive diagnosis of pompholyx. Conclusions Although confirmed intrauterine herpes simplex virus (HSV) infections are very rare, the potentially devastating prognosis and complications as evidenced by our case show that awareness of intrauterine HSV is critical for pediatricians as well as physicians involved in antenatal care. In order to adequately assess, treat and counsel pregnant women, more work is required to detect HSV infection, evaluate possible congenital infection and improve outcomes with fetal and neonatal treatment.","PeriodicalId":9617,"journal":{"name":"Case Reports in Perinatal Medicine","volume":null,"pages":null},"PeriodicalIF":0.1,"publicationDate":"2021-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"74935368","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}
S. Darakci, S. Ertuğrul, S. Tanrıverdi Yılmaz, E. Ünal, İ. Yolbaş, I. Deger
Abstract Objectives Amniotic band syndrome and osteogenesis imperfecta are two distinct diseases that develop due to structural defects of the collagen protein. In our paper, we report the concurrence of these two diseases rarely seen in the newborn period. Case presentation A female infant born at 33rd gestational week was found to have constrictive bands in her right lower extremity and flexion contractures in distal joints of lower and upper extremities due to amniotic bands in postnatal physical examination. While being treated for respiratory difficulty, she was diagnosed with osteogenesis imperfecta and treated with bisphosphonates upon being found to suffer bilateral humeral fractures on the sixth day of life. She received respiratory support with mechanical ventilation due to respiratory tract complications related to osteogenesis imperfecta and died on the 384th day of life. Conclusions One should bear in mind that other collagen tissue diseases may accompany the amniotic band syndrome; this possibility should be definitely pursued if clinical suspicion exists.
{"title":"Infrequent association of two rare diseases: amniotic band syndrome and osteogenesis imperfecta","authors":"S. Darakci, S. Ertuğrul, S. Tanrıverdi Yılmaz, E. Ünal, İ. Yolbaş, I. Deger","doi":"10.1515/crpm-2021-0035","DOIUrl":"https://doi.org/10.1515/crpm-2021-0035","url":null,"abstract":"Abstract Objectives Amniotic band syndrome and osteogenesis imperfecta are two distinct diseases that develop due to structural defects of the collagen protein. In our paper, we report the concurrence of these two diseases rarely seen in the newborn period. Case presentation A female infant born at 33rd gestational week was found to have constrictive bands in her right lower extremity and flexion contractures in distal joints of lower and upper extremities due to amniotic bands in postnatal physical examination. While being treated for respiratory difficulty, she was diagnosed with osteogenesis imperfecta and treated with bisphosphonates upon being found to suffer bilateral humeral fractures on the sixth day of life. She received respiratory support with mechanical ventilation due to respiratory tract complications related to osteogenesis imperfecta and died on the 384th day of life. Conclusions One should bear in mind that other collagen tissue diseases may accompany the amniotic band syndrome; this possibility should be definitely pursued if clinical suspicion exists.","PeriodicalId":9617,"journal":{"name":"Case Reports in Perinatal Medicine","volume":null,"pages":null},"PeriodicalIF":0.1,"publicationDate":"2021-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"90071944","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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