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Biomedical subjects

P Stoutenbeek

Publications and source records attributed to P Stoutenbeek.

At least 19 recordsLinked to original sources
PubMed2004

Neurological outcome of children who were treated for fetal tachycardia complicated by hydrops.

OBJECTIVE: Fetal tachycardia is a condition associated with congestive heart failure and development of fetal hydrops, which may result in neurological morbidity and mortality. The aim of this study was to investigate the long-term outcome of hydropic fetuses. METHODS: This was a retrospective study on cognitive and neurological functioning of 11 infants, aged 6 months to 12 years, who experienced fetal tachycardia complicated by hydrops. RESULTS: Seven fetuses had supraventricular tachycardia (SVT), three had atrial flutter (AF) and one had ventricular tachycardia (VT). Nine fetuses converted to sinus rhythm within a mean time of 8.2 days of presentation; resolution of hydrops was achieved in six of these patients in a mean time of 8.8 days. Mean gestational age (GA) at birth was 35 + 4 weeks. Neonatal cranial ultrasound was normal in seven infants and all but one of these were normal at follow-up: one infant who initially had no abnormalities developed multiple cerebral lesions as a result of a malignant long QT syndrome (LQTS) and died at the age of 2 years. Three infants had periventricular echogenicity (PVE) on neonatal cranial ultrasound, associated with a pseudocyst in one infant. The remaining infant showed a parenchymal hemorrhage of antenatal onset, seen as a porencephalic cyst at birth. One of these infants was normal at follow-up, one died 2 days after birth and two infants had neurological abnormalities at follow-up, consisting of mild hemiplegia with normal cognitive function in one, and a cognitive developmental delay in the other. CONCLUSIONS: In this study, neurological outcome was good in eight out of 11 infants. Initiation of therapy should not be withheld or delayed on the assumption of poor neurological outcome.

Arrhythmias, Cardiac↗
PubMed2004

Treatment of fetal heart block with maternal steroid therapy: case report and review of the literature.

The presence of maternal autoantibodies to SS-A/Ro and/or SS-B/La is associated with the development of fetal heart block. There are data suggesting that maternal treatment with steroids might reverse heart block. We report on a pregnancy in a mother with secondary Sjögren syndrome and systemic lupus erythematosus with presence of autoantibodies to SS-A/Ro and SS-B/La, which was complicated by the development of incomplete fetal heart block. Oral dexamethasone treatment could not prevent progression to complete heart block and was associated with a number of complications.A review of the literature revealed 19 studies (including ours) in which 93 cases of fetal heart block were treated with maternal steroid therapy. Complete heart block proved irreversible in all cases; and of 13 fetuses with incomplete heart block which received maternal steroid therapy, three had a reduction in their degree of block and one reverted to sinus rhythm. Maternal steroid therapy, initiated early in pregnancy and potentially preventing the onset of heart block, did not decrease the incidence of heart block in nine studies with 43 cases. Furthermore, the literature review revealed numerous serious side effects of maternal steroid administration during pregnancy. Data on these potential side effects are lacking in the 28 studies discussed in this review. Maternal dexamethasone therapy to prevent or treat fetal heart block remains, in our opinion, a questionable intervention and can as yet not be recommended in the clinical situation.

Adult↗
PubMed2003

Persistent junctional reciprocating tachycardia in the fetus.

BACKGROUND: Persistent junctional reciprocating tachycardia (PJRT) tends to be a persistent arrhythmia and requires aggressive therapeutic management. Diagnosis and management of this infrequently occurring tachycardia in the fetus at an early stage is of importance for the prevention of congestive heart failure (CHF). METHODS: A retrospective study of four fetuses with supraventricular tachycardia (SVT) of the PJRT type was performed. RESULTS: All had sustained SVT (mean of 228 beats/min) at a mean gestational age of 34 + 5 weeks, with CHF present in two. Three fetuses had prenatal characteristics of PJRT on M-mode echocardiography with a ventriculoatrial (VA)/atrioventricular ratio of > 1 on M-mode echocardiography suggesting a slow conducting accessory pathway. All four fetuses had postnatal confirmation of the diagnosis. Transplacental treatment with flecainide was effective in one patient; sotalol as a single drug or in combination with digoxin was partially effective in the remaining three. Two developed sinus rhythm, with short intermittent periods of tachycardia and decreasing signs of CHF; one case showed a minimal decrease in heart rate. Oral propranolol therapy converted two patients postnatally; in the remaining two patients radiofrequency ablation was performed at the age of 5 months and 6 years. CONCLUSIONS: The characteristics of our prenatal PJRT cases included a sustained heart rate not exceeding 240 beats/min with a long VA interval, the presence of CHF and therapy resistance. Transplacental treatment should be initiated, possibly with a combination of sotalol and digoxin in non-hydropic cases, or flecainide, especially in case of fetal hydrops. Pharmacological therapy is to be preferred postnatally, but radiofrequency ablation seems to be indicated in therapy-resistant cases with CHF, even in the first months of life.

Adult↗
PubMed2002

[Correlation between prenatal ultrasound diagnosis and findings from autopsies or neonatal operations due to structural congenital heart defects; a retrospective analysis].

OBJECTIVE: To determine the contribution of the prenatal ultrasound diagnosis of structural congenital heart defects to the care of patients requiring surgery for their condition in the neonatal period. DESIGN: Retrospective. METHOD: During the period January 1991-June 1999, data were collected from two groups of children. One group had a structural heart defect diagnosed prenatally and the other underwent a neonatal operation due to a heart defect. In the prenatal group, the management and follow-up were analysed, whilst in the surgery group, the extent to which ultrasound investigation had been performed and whether there was a relationship between prenatal diagnosis and the severity of acidosis were analysed. RESULTS: A structural heart defect was found through prenatal ultrasound in 170 children and 64% of these prenatally diagnosed patients died before or immediately after birth, or the pregnancy was aborted. 14% underwent surgery within 31 days post partum. This last category formed an overlap with the surgery group. The surgery group contained 191 patients, who required surgery in the neonatal period, including 24 from the prenatal group. In total, 87% had no prenatal diagnosis. In the group with a prenatal diagnosis pre-operative lactate values were significantly lower. CONCLUSION: The heart defect was only diagnosed prenatally in 13% of the patients from the surgery group, although this would have been technically feasible in 46%. Better prenatal diagnosis of heart disease could contribute to a better counselling of parents in their choice between abortion, conservative treatment or emergency surgery. In the case of emergency surgery, prenatal detection might lead to a decrease in morbidity and mortality.

Acidosis, Lactic↗
PubMed2002

Lactacidosis in the neonate is minimized by prenatal detection of congenital heart disease.

OBJECTIVES: To investigate the impact of prenatal detection of congenital heart disease on preventing severe preoperative lactacidosis. DESIGN: Patients operated upon for congenital heart disease during the first 31 days of life (n = 209) were studied retrospectively, 21 were diagnosed prenatally and 188 patients had not been diagnosed prenatally. Preoperative lactate, pH and base excess were evaluated. RESULTS: Differences were noted in preoperative pH (7.28 +/- 0.03 vs. 7.24 +/- 0.01, P = 0.29), base excess (-5.83 +/- 0.64 vs. -6.93 +/- 0.46 mmol/L, P = 0.10) and lactate (3.05 +/- 0.35 vs. 6.08 +/- 0.45 mmol/L, P < 0.001), indicating a significant difference in blood lactate values in favor of the prenatally diagnosed group. CONCLUSIONS: Prenatal diagnosis of congenital heart disease and the resulting immediate postnatal care prevent lactate increase in the preoperative period of these patients. This may decrease the risk of cerebral damage and result in the patient being in better condition at surgery.

Acidosis, Lactic↗
PubMed2001

[Aneurysm of the V. magna cerebri (vein of Galen), a cause of congestive heart failure in five neonates].

In five neonates (4 girls and 1 boy) with aneurysm of the V. magna cerebri, clinical signs of congestive heart failure were encountered. In three of the neonates the symptoms were detected postnatally and in two prenatally. In one of the prenatally detected cases the aneurysm was treated by embolisation during the first week of life. Upon follow-up at six years of age, this patient's situation was stable but hydrocephalus had led to poor visuomotor development, mild motor retardation and mental retardation. In the other infants, medicinal treatment of the congestive heart failure was discontinued in view of ischaemic brain damage, and these patients subsequently died. Congestive heart failure often develops in neonates with an aneurysm of the V. magna cerebri due to low cerebrovascular resistance. A continuous murmur over the skull is pathognomonic for this condition.

Cerebral Veins↗
PubMed2001

[Protocols for the treatment of supraventricular tachycardias in the fetus].

The protocols mentioned are used by Utrecht University Hospital for the treatment of foetal supraventricular tachycardias. In the case of atrial flutters the pregnant woman is treated with sotalol administered orally and, if no sinus rhythm is obtained nor a reduced ventricular rhythm occurs, subsequently with digoxin. If there is no hydrops foetalis then this is also the treatment regimen for other forms of foetal supraventricular tachycardia. In the case of hydrops foetalis the treatment is more aggressive: digoxin intravenously or flecanide orally; if the rhythm does not convert into a sinus rhythm nor a reduced ventricular rhythm occurs then both of these medications are administered; if that also proves to be insufficient then direct foetal therapy can be considered.

Anti-Arrhythmia Agents↗
PubMed2001

[Congenital infection: diagnostic serology of the mother not always definitive].

In 2 infants, a girl and a boy, congenital viral infection was diagnosed in the neonatal period. The prenatal examination (serologic investigation for Toxoplasma gondii, rubella virus, cytomegalovirus, herpes simplex virus and syphilis (TORCHES)) was negative. In both cases prenatal ultrasonography was abnormal and suggested intrauterine infection. The infants were born with typical symptoms of multisystem disease, known as symptomatic congenital cytomegalovirus infection (jaundice, petechiae, hepatosplenomegaly, intrauterine growth retardation, microcephaly and cerebral calcifications) and congenital rubella syndrome (intrauterine growth retardation, congenital heart disease, cataract, hepatosplenomegaly and cerebral calcifications), respectively. Both had severe cerebral damage. To diagnose severe congenital infection in the first trimester of pregnancy in presence of congenital anomalies in utero there are other possible methods than TORCHES investigation, such as polymerase chain reaction and virus culture in amniotic fluid or in foetal blood obtained by cord puncture.

Adult↗
PubMed2001

[Diagnostic value of prenatal MRI in fetus with intracranial anomalies diagnosed by ultrasonography].

OBJECTIVE: To determine the value of prenatal MRI in the third trimester in the foetus with intracranial lesions established by ultrasonography. DESIGN: Descriptive. METHOD: During a period of one year, prenatal MRI was performed in 12 patients after ultrasonography had revealed intracranial lesions in the foetus. Postnatally, ultrasonography, MRI or postmortem examination of the brain was performed in 11 children. The MRI examination was carried out in a 1.5 Tesla field. Rapid, T2-weighted images were obtained. To immobilize the child, the pregnant women were sedated. RESULTS: In 5 cases, prenatal ultrasonography and prenatal MRI resulted in the same diagnosis. In seven cases, MRI provided more information. Postnatal examination confirmed the prenatal diagnosis in 10 of the 11 cases. In one case, maceration rendered supplementary examination impossible. CONCLUSION: MRI may be regarded as a valuable alternative diagnostic aid if prenatal ultrasonography is incomplete or the findings are uncertain or limited.

Brain↗
PubMed2001

Restrictive dermopathy and fetal behaviour.

We report three siblings from consecutive pregnancies affected with restrictive dermopathy (RD). During the second pregnancy, fetal behavioural development and growth were studied extensively using ultrasound at 1-4 week intervals. Dramatic and sudden changes occurred in fetal body movements and growth but not until the end of the second trimester of pregnancy. Prominent at that time were prolonged periods of fetal quiescence and very low heart rate variability, together with abnormally executed body movements of short duration. Retarded femoral development and jerky abrupt fetal body movements (abnormal movement quality) were already present in the early second trimester of pregnancy. Facial anomalies emerged despite the presence of fetal mouth movements. The clinical features of RD were only partly explained by present knowledge of skin development and the fetal akinesia deformation sequence hypothesis. Quantitative assessment of fetal movements proved to be a poor early marker for antenatal diagnosis of this disorder.

Adult↗
PubMed2001

Prenatal diagnosis of congenital heart disease affects preoperative acidosis in the newborn patient.

OBJECTIVES: Congenital heart disease is the leading cause of death in the first year after birth. Prenatal diagnosis of the disease can optimize the preoperative condition of the patient and may help in the prevention of acidosis. In this retrospective study we compared the occurrence of metabolic acidosis in patients with and without prenatal diagnosis of a congenital heart disease. METHODS: Data of 408 patients who needed an operation for congenital heart disease within 31 days of life were analyzed retrospectively. Arterial blood gases at fixed time intervals and worst blood gas of 81 patients with and 327 patients without a prenatal diagnosis were compared, categorizing the patients on ductus dependency, anticipated univentricular or biventricular repair, and left-sided, right-sided, or no heart obstruction. RESULTS: In the overall group significant differences in lowest pH, lowest base excess, and highest lactate level were found, with metabolic acidosis more common among the patients with a postnatal diagnosis. In the group of patients with ductus-dependent congenital heart disease, the difference between patients receiving a prenatal and those receiving a postnatal diagnosis was more significant than in the group with non-ductus-dependent lesions. Analyzing patients with right-sided, left-sided, and no obstruction separately, significant differences were found in the group with left-sided heart obstruction for lowest pH and base excess and in the group with right-sided heart obstruction for lowest base excess. CONCLUSIONS: Prenatal diagnosis of congenital heart disease minimizes metabolic acidosis in patients with congenital heart disease and may be associated with improved long-term outcome and prevention of cerebral damage among this fragile group of patients, although no significant effect on direct surgical outcome was encountered.

Acidosis↗
PubMed2000

Sotalol in the treatment of fetal dysrhythmias.

BACKGROUND: Fetal tachycardia may cause hydrops fetalis and lead to fetal death. No unanimity of opinion exists regarding the optimum treatment. This study evaluates our experience with transplacental sotalol therapy to treat fetal tachycardias in terms of safety and efficacy. METHODS AND RESULTS: The charts of 21 patients who were treated with sotalol for fetal tachycardia were reviewed. Ten fetuses had atrial flutter (AF), 10 had supraventricular tachycardia (SVT), and 1 had VT. Hydrops fetalis was present in 9 fetuses. Drug treatment was successful in establishing sinus rhythm in 8 of 10 fetuses with AF and in 6 of 10 fetuses with SVT. The mortality rate in this study was 19% (4 of 21 fetuses; 3 had SVT and 1 had AF); 3 deaths occurred just days after the initiation of sotalol therapy, and 1 occurred after a dosage increase. At birth, tachycardia was present in 6 infants. Two patients who converted to sinus rhythm in utero suffered from neurologic pathology postnatally. CONCLUSIONS: Fetal tachycardia is a serious condition in which treatment should be initiated, especially in the presence of hydrops fetalis. The high success rate in fetuses with AF suggests that sotalol should be considered a drug of first choice to treat fetal AF. The low conversion rate and the fact that 3 of the 4 deaths in this study occurred in fetuses with SVT indicate that the risks of sotalol therapy outweigh the benefits in this group and that sotalol should, therefore, be limited in the treatment of fetal SVT.

Anti-Arrhythmia Agents↗
PubMed2000

Atrial flutter in the perinatal age group: diagnosis, management and outcome.

OBJECTIVES: The aim of this retrospective study was to evaluate perinatal atrial flutter (AF) and the efficacy of maternally administered antiarrhythmic agents, postpartum management and outcome. BACKGROUND: Perinatal AF is a potentially lethal arrhythmia, and management of this disorder is difficult and controversial. METHODS: Forty-five patients with documented AF were studied retrospectively. RESULTS: Atrial flutter was diagnosed prenatally in 44 fetuses and immediately postnatally in 1 neonate. Fetal hydrops was seen in 20 patients; 17 received maternal therapy, 2 were delivered and 1 was not treated because it had a severe nontreatable cardiac malformation. In the nonhydropic group of 24 patients, 18 were treated and the remaining 6 were delivered immediately. In the hydropic group, 10 received single-drug therapy (digoxin or sotalol) and 7 received multidrug therapy. In the nonhydropic group, 13 received a single drug (digoxin or sotalol) and 5 received multiple drugs. One patient with rapid 1:1 atrioventricular conduction (heart rate 480 beats/min) died in utero and another died due to a combination of severe hydrops because of the AF, sotalol medication, stenosis of the venous duct and hypoplastic placenta. Of the 43 live-born infants, 12 were in AF at birth. Electrical cardioversion was successful in eight of nine patients. No recurrences in AF have occurred beyond the neonatal period. Four patients with fetal flutter and hydrops showed significant neurological pathology immediately after birth. CONCLUSIONS: Fetal AF is a serious and threatening rhythm disorder, particularly when it causes hydrops, it may be associated with fetal death or neurological damage. Treatment is required and primarily aimed at reaching an adequate ventricular rate and preferably conversion to sinus rhythm. Digoxin failed in prevention of recurrence at time of delivery in a quarter of our patients, whereas with sotalol no recurrence of AF has been reported, suggesting that class III agents may be the future therapy. Once fetuses with AF survive without neurological pathology, their future is good and prophylaxis beyond the neonatal period is unnecessary.

Anti-Arrhythmia Agents↗
PubMed1999

[Third circulation: twin transfusion syndrome].

The estimated incidence in the Netherlands of serious previable twin-to-twin transfusion syndrome is 50-100 cases per year. The polyhydramnion-oligohydramnion sequence is the most prominent feature. Prognosis without treatment is dismal: previable preterm birth. Risk of serious morbidity in the surviving twin in case of foetal demise of the other is impressive. Once the diagnosis of monochorionicity has been made in the first trimester, detailed ultrasound examination is mandatory for early recognition of the development of the syndrome, as therapeutic options exist. Therapeutic options include piercing of the intertwin membrane, repeated amniocenteses or laser occlusion of the chorionic vasculature. In seven Dutch centres over three years time 61 cases were identified: 36 needed intervention; 63 (of the 122) children survived, 10 with longterm morbidity.

Amniocentesis↗
PubMed1999

Isochromosome 1q as the sole chromosomal abnormality in two fetal teratomas. Possible trisomic or tetrasomic zygote rescue in fetal teratoma with an additional isochromosome 1q.

An isochromosome of the long arm of chromosome 1 leading to tetrasomy 1q was detected as the sole chromosomal aberration in two cases of fetal teratoma arising from the oral cavity. This type of teratoma is extremely rare and has seldom been investigated cytogenetically. Studies of DNA markers in the tumor, normal fetal skin, and parental cells demonstrated that in both cases the additional 1q material was of maternal origin. In one of the patients, the teratoma had maternal 1q marker alleles that were not found in the fetal body cells. This implies that the tumor was not derived in a direct way from the fetal body tissue; instead, the chromosomally-normal fetus might be the result of some trisomic or tetrasomic zygote rescue mechanism.

Alleles↗
PubMed1999

Neurological morbidity after fetal supraventricular tachyarrhythmia.

BACKGROUND: Fetal tachyarrhythmia is a well-documented entity which, in the absence of pharmacological intervention, may lead to congestive heart failure, fetal hydrops and eventually fetal demise. The success rate of the implemented treatment is generally measured by survival and achievement of control of the arrhythmia. We report on the occurrence of associated cerebral damage in three patients with fetal tachycardia. METHODS: We describe three patients with a history of fetal supraventricular tachyarrhythmia who developed cerebral complications in utero. RESULTS: Two patients had cerebral hypoxic-ischemic lesions and one had hemorrhagic lesions present at birth. They had developed severe congestive heart failure and fetal hydrops secondary to fetal tachyarrhythmia, and there were no other obvious causes for the cerebral pathology. Two of these patients were referred to us antenatally. Therapy was instituted and resulted in control of the tachycardia and resolution of hydrops. The third patient was referred to our clinic shortly after birth because of severe circulatory problems secondary to fetal tachyarrhythmia. CONCLUSION: From these observations, we believe that a fetus with tachyarrhythmia and subsequent hydrops is at increased risk for the development of cerebral complications, due to the circulatory disturbances and sudden changes in heart rate which may lead to fluctuations in cerebral perfusion. This would imply that it is of the utmost importance to aim at immediate and complete control of the heart rate in the treatment of fetal tachyarrhythmia.

Adult↗