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Papers by Enrico Lopriore

Research paper thumbnail of Cardiac arrhythmias associated with umbilical venous catheterisation in neonates

Research paper thumbnail of Poor accuracy of methods currently used to determine umbilical catheter insertion length

Research paper thumbnail of Revised formula to determine the insertion length of umbilical vein catheters

European Journal of Pediatrics, 2013

The method of Shukla is commonly used to predict the insertion length of umbilical vein catheters... more The method of Shukla is commonly used to predict the insertion length of umbilical vein catheters (UVCs) but often leads to over-insertion. Malposition of UVCs can lead to complications. In this study, we compared the formula of Shukla, i.e., (3 x birthweight in kg + 9) / 2 + 1 cm with a revised formula, i.e., (3 x birthweight in kg + 9) / 2 cm in determining the insertion length of UVCs. A cohort where the revised formula was used for UVC placement (revised group) was compared with a historical cohort using the conventional formula (Shukla group). We evaluated the position of UVCs stated as the corresponding vertebra level with a radiograph of the infant's chest and abdomen immediately after insertion in both groups. Positioning of the catheter tip above the ninth or below the tenth thoracic vertebra was considered too high or too low, respectively. Median position of 93 UVCs placed according to Shukla was lower (seventh thoracic vertebra, interquartile range (IQR) 6-9) when compared to 92 UVCs placed according to the revised formula (eighth thoracic vertebra (IQR 7-9)). UVCs were more often over-inserted using the Shukla formula (73%) when compared to the revised formula (54%). One UVC in the Shukla group (1%) and two UVCs in the revised group (2%) were placed too low (p = NS). The revised formula reduces the rate of over-insertion of UVCs without increasing the rate of inadequate lower positioning.

Research paper thumbnail of Residual anastomoses after fetoscopic laser surgery in twin-to- twin transfusion syndrome: frequency, associated risks and outcome

Research paper thumbnail of Long-term neurodevelopmental outcome in twin-to-twin transfusion syndrome treated with fetoscopic laser surgery

Research paper thumbnail of Fetoscopic laser surgery in 100 pregnancies with severe twin-to-twin transfusion syndrome in the Netherlands

Research paper thumbnail of The Ages and Stages Questionnaire and Neurodevelopmental Impairment in Two-Year-Old Preterm-Born Children

PLOS ONE, 2015

To test the ability of the Ages and Stages Questionnaire, Third Edition (ASQ3) to help identify o... more To test the ability of the Ages and Stages Questionnaire, Third Edition (ASQ3) to help identify or exclude neurodevelopmental impairment (NDI) in very preterm-born children at the corrected age of two. We studied the test results of 224 children, born at <32 postmenstrual weeks, who had scores on ASQ3 and Bayley Scales of Infant and Toddler Development, Third Edition (BSIDIII) and neurological examination at 22-26 months' corrected age. We defined NDI as a score of <70 on the cognitive-or motor composite scale of BSIDIII, or impairment on neurological examination or audiovisual screening. We compared NDI with abnormal ASQ3 scores, i.e., < -2SDs on any domain, and with ASQ3 total scores. To correct for possible overestimation of BSIDIII, we also analyzed the adjusted BSIDIII thresholds for NDI, i.e., scores <80 and <85. We found 61 (27%) children with abnormal ASQ3 scores, and 10 (4.5%) children who had NDI with original BSIDIII thresholds (<70). Twelve children had NDI at BSIDIII thresholds at <80, and 15 had <85. None of the 163 (73%) children who passed ASQ3 had NDI. The sensitivity of ASQ3 to detect NDI was excellent (100%), its specificity was acceptable (76%), and its negative predictive value (NPV) was 100%. Sensitivity and NPV remained high with the adjusted BSIDIII thresholds. The Ages and Stages Questionnaire is a simple, valid and cost-effective screening tool to help identify and exclude NDI in very preterm-born children at the corrected age of two years.

Research paper thumbnail of Middle cerebral artery peak systolic velocity to predict fetal hemoglobin levels in Twin Anemia Polycythemia Sequence

Ultrasound in obstetrics & gynecology : the official journal of the International Society of Ultrasound in Obstetrics and Gynecology, Jan 10, 2015

Our aim was to evaluate the diagnostic accuracy of middle cerebral artery peak systolic velocity ... more Our aim was to evaluate the diagnostic accuracy of middle cerebral artery peak systolic velocity (MCA-PSV) Doppler measurements in twin anemia polycythemia sequence (TAPS). In a consecutive cohort of monochorionic twin pregnancies with TAPS between 2005 and 2013 in three European fetal therapy centers, the accuracy to predict anemia and polycythemia of MCA-PSV measured just prior to fetal hemoglobin (Hb) measurement by fetal or cord blood sampling was assessed using 2x2 tables. A total of 116 measurements (74 in donors and 42 in recipients) from 43 TAPS cases could be used for analysis. MCA-PSV multiples of the mean (MoM) values correlated well with Hb levels (R = -0.86 P < 0.001). The sensitivity of the MCA-PSV > 1.5 MoM to predict severe anemia (Hb deficit >5 standard deviations (SD)) in TAPS donors was 94% (95%CI 85-98%), specificity 74% (95%CI 62-83%), positive predictive value 76% (95%CI 65-85%), negative predictive value 94% (95%CI 83-98%). The sensitivity of MCA-PSV ...

Research paper thumbnail of Perinatal mortality and mode of delivery in monochorionic diamniotic twin pregnancies >/= 32 weeks of gestation: a multicentre retrospective cohort study

Bjog-an International Journal of Obstetrics and Gynaecology, 2011

OBJECTIVE: To study perinatal mortality rates in a cohort of 465 monochorionic (MC) twins without... more OBJECTIVE: To study perinatal mortality rates in a cohort of 465 monochorionic (MC) twins without twin-twin transfusion syndrome (TTS) born at 32 weeks of gestation or later since reported interauterine fetal death (IUFD) rates >32 weeks of gestations in the literature vary, leading to varying recommendations on the optimal timing of delivery, and to investigate the relation between perinatal mortality

Research paper thumbnail of OP09.07: TTTS1 protocol: a cluster randomized controlled trial comparing a conservative management and primary laser surgery in stage 1 TTTS

Ultrasound in Obstetrics & Gynecology, 2009

Research paper thumbnail of Timely diagnosis of twin-to-twin transfusion syndrome in monochorionic twin pregnancies by biweekly sonography combined with patient instruction to report onset of symptoms

Ultrasound in obstetrics & gynecology : the official journal of the International Society of Ultrasound in Obstetrics and Gynecology, 2006

To assess the value of serial ultrasound examinations together with patient instructions to repor... more To assess the value of serial ultrasound examinations together with patient instructions to report the onset of symptoms in achieving timely detection of twin-to-twin transfusion syndrome (TTTS) in a cohort of monochorionic diamniotic twin pregnancies, and to evaluate sonographic TTTS predictors. Timely detection of TTTS was defined as diagnosis before severe complications of TTTS occurred, such as preterm prelabor rupture of membranes, very preterm delivery (24-32 weeks of pregnancy), fetal hydrops, or intrauterine fetal death. During a 2-year period, a prospective series of 23 monochorionic twin pregnancies was monitored from the first trimester until delivery. At least every 2 weeks we performed ultrasound and Doppler measurements (nuchal translucency thickness, presence of membrane folding, estimated fetal weight, deepest vertical pocket, bladder filling, and Doppler waveforms of the umbilical artery, ductus venosus and umbilical vein). Measurements of TTTS cases were compared w...

Research paper thumbnail of Twin pregnancies with two separate placental masses can still be monochorionic and have vascular anastomoses

American journal of obstetrics and gynecology, 2006

This study was undertaken to report the occurrence of bipartite monochorionic twin placentas. Exa... more This study was undertaken to report the occurrence of bipartite monochorionic twin placentas. Examination of 109 monochorionic placentas delivered at our institution between June 2002 and June 2005 was performed. Placental characteristics on prenatal ultrasound were studied, including single or double appearance and type of intertwin membrane-placental junction ("T" sign or lambda sign). Monochorionicity was confirmed by postnatal histologic confirmation (diamniotic intertwin membrane without chorionic tissue within the dividing septum). Bipartition was diagnosed when 2 separate placental masses attached by membranes were identified. Of the 109 monochorionic placentas, 3 were composed of 2 separate placental masses. Prenatal ultrasound examination showed 2 separate placental masses in each case. Monochorionicity was suspected on prenatal ultrasound because of the presence of "T" sign in 2 cases and twin-to-twin transfusion syndrome (TTTS) in another case. Microsc...

Research paper thumbnail of Residual anastomoses in twin-twin transfusion syndrome after laser: the Solomon randomized trial

American Journal of Obstetrics and Gynecology, 2014

Research paper thumbnail of Patient controlled analgesia with remifentanil versus epidural analgesia in labour: randomised multicentre equivalence trial

BMJ (Clinical research ed.), 2015

To determine women's satisfaction with pain relief using patient controlled analgesia with re... more To determine women's satisfaction with pain relief using patient controlled analgesia with remifentanil compared with epidural analgesia during labour. Multicentre randomised controlled equivalence trial. 15 hospitals in the Netherlands. Women with an intermediate to high obstetric risk with an intention to deliver vaginally. To exclude a clinically relevant difference in satisfaction with pain relief of more than 10%, we needed to include 1136 women. Because of missing values for satisfaction this number was increased to 1400 before any analysis. We used multiple imputation to correct for missing data. Before the onset of active labour consenting women were randomised to a pain relief strategy with patient controlled remifentanil or epidural analgesia if they requested pain relief during labour. Primary outcome was satisfaction with pain relief, measured hourly on a visual analogue scale and expressed as area under the curve (AUC), thus providing a time weighted measure of tota...

Research paper thumbnail of Neonatal carnitine palmitoyltransferase II deficiency: failure of treatment despite prolonged survival

Carnitine palmitoyltransferase (CPT) deficiencies are disorders of mitochondrial fatty acid oxida... more Carnitine palmitoyltransferase (CPT) deficiencies are disorders of mitochondrial fatty acid oxidation (FAO). In fatty acid oxidation, long-chain fatty acids need the carnitine cycle to be transported from the cytosol to the mitochondria. In CPT II deficiency, long-chain acylcarnitines cannot be metabolised to carnitine and acyl-CoA, leading to accumulation of toxic long-chain acylcarnitines. Three clinical presentations of CPT II deficiency have been identified: the adult form, the infantile form and the neonatal form. The neonatal form of CPT II is the most severe and all reported patients died within a few days to 6 weeks after birth. The first case of a patient with neonatal CPT II deficiency surviving beyond the neonatal period is described. Unfortunately, the infant died at the age of 6 months due to untreatable cardiac arrhythmias.

Research paper thumbnail of Top-up transfusions in neonates with Rh hemolytic disease in relation to exchange transfusions

Research paper thumbnail of Successful ultrasound-guided laser treatment of fetal hydrops caused by pulmonary sequestration

Ultrasound in Obstetrics and Gynecology, 2007

Research paper thumbnail of Rhesus haemolytic disease of the newborn: Postnatal management, associated morbidity and long-term outcome

Seminars in Fetal and Neonatal Medicine, 2008

Rhesus haemolytic disease of the newborn can lead to complications such as hyperbilirubinaemia, k... more Rhesus haemolytic disease of the newborn can lead to complications such as hyperbilirubinaemia, kernicterus and anaemia. Postnatal management consists mainly of intensive phototherapy, exchange transfusion and blood transfusion. During the last decades, significant progress in prenatal care strategies for patients with Rhesus haemolytic disease has occurred. New prenatal management options have led to a remarkable reduction in perinatal mortality. As a result of the increase in perinatal survival, attention is now shifting towards short-term and long-term morbidity. This review focuses on the management of neonatal and paediatric complications associated with Rhesus haemolytic disease, discusses postnatal treatment options and summarizes the results of studies on short-term and long-term outcome.

Research paper thumbnail of Haemoglobin differences at birth in monochorionic twins without chronic twin-to-twin transfusion syndrome

Research paper thumbnail of Deep-hidden anastomoses in monochorionic twin placentae are harmless

Prenatal Diagnosis, 2007

Our objective was to identify the clinical consequences of deep-hidden anastomoses that occur und... more Our objective was to identify the clinical consequences of deep-hidden anastomoses that occur underneath the placental surface. Twelve placentae that underwent intrauterine laser ablation of placental anastomoses for twin-twin transfusion syndrome (TTTS) and 14 non-TTTS controls were investigated for deep-hidden anastomoses. Additionally, we investigated the inter-twin haemoglobin differences as an indicator for fetofetal transfusion. Placentae were divided into four groups: TTTS placentae without residual chorionic-plate anastomoses without deep-hidden anastomoses (group 1) and with deep-hidden anastomoses (group 2), and non-TTTS placentae with chorionic-plate anastomoses without deep-hidden anastomoses (group 3) and with deep-hidden anastomoses (group 4). Deep-hidden anastomoses were identified in 58% (7/12) of the TTTS placentae after laser surgery and in 64% (9/14) of the non-TTTS placentae. Groups 1 and 2 had equal inter-twin haemoglobin differences: medians 1.4 and 1.2 gr/dL, respectively (p = 0.48). In group 3, the median inter-twin haemoglobin difference without deep-hidden anastomoses was 2.6 gr/dL (group 3) and with deep-hidden anastomoses (group 4) it was 5.1 gr/dL (p = 0.26). Both comparisons imply that deep-hidden anastomoses did not cause any additional increase in Hb difference. In conclusion, haematological and additional hemodynamical analysis show that deep-hidden anastomoses are likely to occur without any clinical consequences.

Research paper thumbnail of Cardiac arrhythmias associated with umbilical venous catheterisation in neonates

Research paper thumbnail of Poor accuracy of methods currently used to determine umbilical catheter insertion length

Research paper thumbnail of Revised formula to determine the insertion length of umbilical vein catheters

European Journal of Pediatrics, 2013

The method of Shukla is commonly used to predict the insertion length of umbilical vein catheters... more The method of Shukla is commonly used to predict the insertion length of umbilical vein catheters (UVCs) but often leads to over-insertion. Malposition of UVCs can lead to complications. In this study, we compared the formula of Shukla, i.e., (3 x birthweight in kg + 9) / 2 + 1 cm with a revised formula, i.e., (3 x birthweight in kg + 9) / 2 cm in determining the insertion length of UVCs. A cohort where the revised formula was used for UVC placement (revised group) was compared with a historical cohort using the conventional formula (Shukla group). We evaluated the position of UVCs stated as the corresponding vertebra level with a radiograph of the infant&amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;#39;s chest and abdomen immediately after insertion in both groups. Positioning of the catheter tip above the ninth or below the tenth thoracic vertebra was considered too high or too low, respectively. Median position of 93 UVCs placed according to Shukla was lower (seventh thoracic vertebra, interquartile range (IQR) 6-9) when compared to 92 UVCs placed according to the revised formula (eighth thoracic vertebra (IQR 7-9)). UVCs were more often over-inserted using the Shukla formula (73%) when compared to the revised formula (54%). One UVC in the Shukla group (1%) and two UVCs in the revised group (2%) were placed too low (p = NS). The revised formula reduces the rate of over-insertion of UVCs without increasing the rate of inadequate lower positioning.

Research paper thumbnail of Residual anastomoses after fetoscopic laser surgery in twin-to- twin transfusion syndrome: frequency, associated risks and outcome

Research paper thumbnail of Long-term neurodevelopmental outcome in twin-to-twin transfusion syndrome treated with fetoscopic laser surgery

Research paper thumbnail of Fetoscopic laser surgery in 100 pregnancies with severe twin-to-twin transfusion syndrome in the Netherlands

Research paper thumbnail of The Ages and Stages Questionnaire and Neurodevelopmental Impairment in Two-Year-Old Preterm-Born Children

PLOS ONE, 2015

To test the ability of the Ages and Stages Questionnaire, Third Edition (ASQ3) to help identify o... more To test the ability of the Ages and Stages Questionnaire, Third Edition (ASQ3) to help identify or exclude neurodevelopmental impairment (NDI) in very preterm-born children at the corrected age of two. We studied the test results of 224 children, born at &amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;lt;32 postmenstrual weeks, who had scores on ASQ3 and Bayley Scales of Infant and Toddler Development, Third Edition (BSIDIII) and neurological examination at 22-26 months&amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;#39; corrected age. We defined NDI as a score of &amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;lt;70 on the cognitive-or motor composite scale of BSIDIII, or impairment on neurological examination or audiovisual screening. We compared NDI with abnormal ASQ3 scores, i.e., &amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;lt; -2SDs on any domain, and with ASQ3 total scores. To correct for possible overestimation of BSIDIII, we also analyzed the adjusted BSIDIII thresholds for NDI, i.e., scores &amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;lt;80 and &amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;lt;85. We found 61 (27%) children with abnormal ASQ3 scores, and 10 (4.5%) children who had NDI with original BSIDIII thresholds (&amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;lt;70). Twelve children had NDI at BSIDIII thresholds at &amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;lt;80, and 15 had &amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;lt;85. None of the 163 (73%) children who passed ASQ3 had NDI. The sensitivity of ASQ3 to detect NDI was excellent (100%), its specificity was acceptable (76%), and its negative predictive value (NPV) was 100%. Sensitivity and NPV remained high with the adjusted BSIDIII thresholds. The Ages and Stages Questionnaire is a simple, valid and cost-effective screening tool to help identify and exclude NDI in very preterm-born children at the corrected age of two years.

Research paper thumbnail of Middle cerebral artery peak systolic velocity to predict fetal hemoglobin levels in Twin Anemia Polycythemia Sequence

Ultrasound in obstetrics & gynecology : the official journal of the International Society of Ultrasound in Obstetrics and Gynecology, Jan 10, 2015

Our aim was to evaluate the diagnostic accuracy of middle cerebral artery peak systolic velocity ... more Our aim was to evaluate the diagnostic accuracy of middle cerebral artery peak systolic velocity (MCA-PSV) Doppler measurements in twin anemia polycythemia sequence (TAPS). In a consecutive cohort of monochorionic twin pregnancies with TAPS between 2005 and 2013 in three European fetal therapy centers, the accuracy to predict anemia and polycythemia of MCA-PSV measured just prior to fetal hemoglobin (Hb) measurement by fetal or cord blood sampling was assessed using 2x2 tables. A total of 116 measurements (74 in donors and 42 in recipients) from 43 TAPS cases could be used for analysis. MCA-PSV multiples of the mean (MoM) values correlated well with Hb levels (R = -0.86 P < 0.001). The sensitivity of the MCA-PSV > 1.5 MoM to predict severe anemia (Hb deficit >5 standard deviations (SD)) in TAPS donors was 94% (95%CI 85-98%), specificity 74% (95%CI 62-83%), positive predictive value 76% (95%CI 65-85%), negative predictive value 94% (95%CI 83-98%). The sensitivity of MCA-PSV ...

Research paper thumbnail of Perinatal mortality and mode of delivery in monochorionic diamniotic twin pregnancies >/= 32 weeks of gestation: a multicentre retrospective cohort study

Bjog-an International Journal of Obstetrics and Gynaecology, 2011

OBJECTIVE: To study perinatal mortality rates in a cohort of 465 monochorionic (MC) twins without... more OBJECTIVE: To study perinatal mortality rates in a cohort of 465 monochorionic (MC) twins without twin-twin transfusion syndrome (TTS) born at 32 weeks of gestation or later since reported interauterine fetal death (IUFD) rates >32 weeks of gestations in the literature vary, leading to varying recommendations on the optimal timing of delivery, and to investigate the relation between perinatal mortality

Research paper thumbnail of OP09.07: TTTS1 protocol: a cluster randomized controlled trial comparing a conservative management and primary laser surgery in stage 1 TTTS

Ultrasound in Obstetrics & Gynecology, 2009

Research paper thumbnail of Timely diagnosis of twin-to-twin transfusion syndrome in monochorionic twin pregnancies by biweekly sonography combined with patient instruction to report onset of symptoms

Ultrasound in obstetrics & gynecology : the official journal of the International Society of Ultrasound in Obstetrics and Gynecology, 2006

To assess the value of serial ultrasound examinations together with patient instructions to repor... more To assess the value of serial ultrasound examinations together with patient instructions to report the onset of symptoms in achieving timely detection of twin-to-twin transfusion syndrome (TTTS) in a cohort of monochorionic diamniotic twin pregnancies, and to evaluate sonographic TTTS predictors. Timely detection of TTTS was defined as diagnosis before severe complications of TTTS occurred, such as preterm prelabor rupture of membranes, very preterm delivery (24-32 weeks of pregnancy), fetal hydrops, or intrauterine fetal death. During a 2-year period, a prospective series of 23 monochorionic twin pregnancies was monitored from the first trimester until delivery. At least every 2 weeks we performed ultrasound and Doppler measurements (nuchal translucency thickness, presence of membrane folding, estimated fetal weight, deepest vertical pocket, bladder filling, and Doppler waveforms of the umbilical artery, ductus venosus and umbilical vein). Measurements of TTTS cases were compared w...

Research paper thumbnail of Twin pregnancies with two separate placental masses can still be monochorionic and have vascular anastomoses

American journal of obstetrics and gynecology, 2006

This study was undertaken to report the occurrence of bipartite monochorionic twin placentas. Exa... more This study was undertaken to report the occurrence of bipartite monochorionic twin placentas. Examination of 109 monochorionic placentas delivered at our institution between June 2002 and June 2005 was performed. Placental characteristics on prenatal ultrasound were studied, including single or double appearance and type of intertwin membrane-placental junction ("T" sign or lambda sign). Monochorionicity was confirmed by postnatal histologic confirmation (diamniotic intertwin membrane without chorionic tissue within the dividing septum). Bipartition was diagnosed when 2 separate placental masses attached by membranes were identified. Of the 109 monochorionic placentas, 3 were composed of 2 separate placental masses. Prenatal ultrasound examination showed 2 separate placental masses in each case. Monochorionicity was suspected on prenatal ultrasound because of the presence of "T" sign in 2 cases and twin-to-twin transfusion syndrome (TTTS) in another case. Microsc...

Research paper thumbnail of Residual anastomoses in twin-twin transfusion syndrome after laser: the Solomon randomized trial

American Journal of Obstetrics and Gynecology, 2014

Research paper thumbnail of Patient controlled analgesia with remifentanil versus epidural analgesia in labour: randomised multicentre equivalence trial

BMJ (Clinical research ed.), 2015

To determine women's satisfaction with pain relief using patient controlled analgesia with re... more To determine women's satisfaction with pain relief using patient controlled analgesia with remifentanil compared with epidural analgesia during labour. Multicentre randomised controlled equivalence trial. 15 hospitals in the Netherlands. Women with an intermediate to high obstetric risk with an intention to deliver vaginally. To exclude a clinically relevant difference in satisfaction with pain relief of more than 10%, we needed to include 1136 women. Because of missing values for satisfaction this number was increased to 1400 before any analysis. We used multiple imputation to correct for missing data. Before the onset of active labour consenting women were randomised to a pain relief strategy with patient controlled remifentanil or epidural analgesia if they requested pain relief during labour. Primary outcome was satisfaction with pain relief, measured hourly on a visual analogue scale and expressed as area under the curve (AUC), thus providing a time weighted measure of tota...

Research paper thumbnail of Neonatal carnitine palmitoyltransferase II deficiency: failure of treatment despite prolonged survival

Carnitine palmitoyltransferase (CPT) deficiencies are disorders of mitochondrial fatty acid oxida... more Carnitine palmitoyltransferase (CPT) deficiencies are disorders of mitochondrial fatty acid oxidation (FAO). In fatty acid oxidation, long-chain fatty acids need the carnitine cycle to be transported from the cytosol to the mitochondria. In CPT II deficiency, long-chain acylcarnitines cannot be metabolised to carnitine and acyl-CoA, leading to accumulation of toxic long-chain acylcarnitines. Three clinical presentations of CPT II deficiency have been identified: the adult form, the infantile form and the neonatal form. The neonatal form of CPT II is the most severe and all reported patients died within a few days to 6 weeks after birth. The first case of a patient with neonatal CPT II deficiency surviving beyond the neonatal period is described. Unfortunately, the infant died at the age of 6 months due to untreatable cardiac arrhythmias.

Research paper thumbnail of Top-up transfusions in neonates with Rh hemolytic disease in relation to exchange transfusions

Research paper thumbnail of Successful ultrasound-guided laser treatment of fetal hydrops caused by pulmonary sequestration

Ultrasound in Obstetrics and Gynecology, 2007

Research paper thumbnail of Rhesus haemolytic disease of the newborn: Postnatal management, associated morbidity and long-term outcome

Seminars in Fetal and Neonatal Medicine, 2008

Rhesus haemolytic disease of the newborn can lead to complications such as hyperbilirubinaemia, k... more Rhesus haemolytic disease of the newborn can lead to complications such as hyperbilirubinaemia, kernicterus and anaemia. Postnatal management consists mainly of intensive phototherapy, exchange transfusion and blood transfusion. During the last decades, significant progress in prenatal care strategies for patients with Rhesus haemolytic disease has occurred. New prenatal management options have led to a remarkable reduction in perinatal mortality. As a result of the increase in perinatal survival, attention is now shifting towards short-term and long-term morbidity. This review focuses on the management of neonatal and paediatric complications associated with Rhesus haemolytic disease, discusses postnatal treatment options and summarizes the results of studies on short-term and long-term outcome.

Research paper thumbnail of Haemoglobin differences at birth in monochorionic twins without chronic twin-to-twin transfusion syndrome

Research paper thumbnail of Deep-hidden anastomoses in monochorionic twin placentae are harmless

Prenatal Diagnosis, 2007

Our objective was to identify the clinical consequences of deep-hidden anastomoses that occur und... more Our objective was to identify the clinical consequences of deep-hidden anastomoses that occur underneath the placental surface. Twelve placentae that underwent intrauterine laser ablation of placental anastomoses for twin-twin transfusion syndrome (TTTS) and 14 non-TTTS controls were investigated for deep-hidden anastomoses. Additionally, we investigated the inter-twin haemoglobin differences as an indicator for fetofetal transfusion. Placentae were divided into four groups: TTTS placentae without residual chorionic-plate anastomoses without deep-hidden anastomoses (group 1) and with deep-hidden anastomoses (group 2), and non-TTTS placentae with chorionic-plate anastomoses without deep-hidden anastomoses (group 3) and with deep-hidden anastomoses (group 4). Deep-hidden anastomoses were identified in 58% (7/12) of the TTTS placentae after laser surgery and in 64% (9/14) of the non-TTTS placentae. Groups 1 and 2 had equal inter-twin haemoglobin differences: medians 1.4 and 1.2 gr/dL, respectively (p = 0.48). In group 3, the median inter-twin haemoglobin difference without deep-hidden anastomoses was 2.6 gr/dL (group 3) and with deep-hidden anastomoses (group 4) it was 5.1 gr/dL (p = 0.26). Both comparisons imply that deep-hidden anastomoses did not cause any additional increase in Hb difference. In conclusion, haematological and additional hemodynamical analysis show that deep-hidden anastomoses are likely to occur without any clinical consequences.