1Department of Gynaecology and Obstetrics and 2Department of Paediatrics and Neonatology, Academic Hospital Maastricht,. Maastricht, The Netherlands.
EuropeanJournalof
Eur J Pediatr (1990) 149 : 261-262
Pediatrics
9 Springer-Verlag 1990
Congenital cytomegalovirus infection in a twin pregnancy: a case report J. J. Duvekot I, B. A. M. Theewes 2, J. M. Wesdorp 2, F. J. M. E. Roumen 3, and P. X. J. M. Bouckaert 3 1Department of Gynaecology and Obstetrics and 2Department of Paediatrics and Neonatology, Academic Hospital Maastricht, Maastricht, The Netherlands 3Department of Gynaecology and Obstetrics, Sint Elisabeth Clinic, 6418 PB Heerlen, The Netherlands
Abstract. A case of congenital cytomegalovirus infection in a twin pregnancy is described, causing neonatal death in one of the infants whereas the other survived without major complications. The possible mechanisms involved are discussed. Key words: Cytomegalovirus - Infection - Congenital - Twins
Introduction Cytomegalovirus (CMV) infection is endemic throughout the world. In over 90% of cases the course of the infection is asymptomatic [5]. In pregnancy, CMV infection is considered the leading cause of congenital viral infection with an incidence of approximately 1% of all live births in the western world, a figure that tends to be higher in lower socio-economic classes [1, 10]. In contrast to rubella both primary and recurrent CMV infection may cause congenital CMV infection [2, 6, 9]. The transmission rate of primary CMV infection is estimated to be 31% -45% [3, 10]. The transmission rate in recurrent infections is significantly lower [1, 9]. Primary CMV infection causes more serious congenital CMV infection than recurrent infection [1, 5]. The mean birth weight of children with congenital CMV infection is significantly lower than that of unaffected children [7]. Especially small-for-gestational-age infants tend to be more severely affected than those with a normal weight [6]. The question arises if this last finding can be explained because of the greater susceptibility of the smallfor-gestational-age infant for intra-uterine infections or that the fetus becomes secondarily growth retarded as a result of an intra-uterine infection.
Case report After a period of a primary infertility, pregnancy was accomplished in a 28-year-old woman, after clomiphene therapy and donor insemination. In the 1st trimester a viable twin pregnancy was detected by ultrasound examination. A t 30 weeks gestation the patient was hospitalized because of discordant growth between both fetuses as diagnosed by ultrasound using the biparietal diameter and the fetal abdominal circumference. No congenital anomalies were seen. A t a gestational age of 32 weeks and 4 days the fetal heart rate pattern of the smallest fetus demonstrated uniform decelerations and a loss of beat-to-beat variation and accelerations. In order to assess Offprint requests to: P. X. J. M. Bouckaert
fetal lung maturity, amniocentesis was performed. Amniotic fluid was sampled from the fluid compartment of the largest fetus. The lecithin/sphingomyelin ratio was 4:1. Amniotic fluid collection from the gestational sac of the small fetus was technically impossible. Two days later the pregnancy was terminated by emergency caesarean section because of increasing signs of fetal distress in the smallest fetus. Two male infants were delivered. The first-born infant had a birth weight of 1190 g (percentile < 2.3 [4]). Apgar-scores were 3, 7 and 9 after 1, 5 and 10 min respectively. Arterial cord blood p H was 6.97. The second-born infant weighed 1670g (percentile 15 [4]). Apgar-scores were 7 and 10 after 1 and 5min respectively. Arterial cord blood p H was 7.27. The placenta was dichorionic, diamniotic and demonstrated no clear microscopic abnormalities, especially no signs of intra-uterine infection. The smallest infant was immediately transferred to a neonatal intensive care unit. The infant was artificially ventilated because of respiratory insufficiency [4]. The child was tachypnoeic and showed costal retractions, rgles were heard over both lungs. Heart rate and blood pressure were normal and no heart murmurs were heard. The liver was palpable 2 cm below the costal margin. Laboratory investigations showed a blood haemoglobin level of 6.6mmol/1 (11.55g/dl) and a normal white cell count, differentiation and platelet count. Chest X-ray demonstrated cardiomegaly and normal lung fields without signs of respiratory distress syndrome. CMV was cultured from the infant's urine and C M V - I g M antibodies were shown in fetal cord serum. No CMV antibodies could be detected in the CSF. Shortly after admission respiration was stabilized by means of continuous nasal positive pressure respiration. After 2 days the infant developed haemorrhagic diathesis. Cranial ultrasound revealed a grade 4 intra-ventricular haemorrhage and convulsions developed. On the 3rd day after birth the infant showed hyperbilirubinaemia which required phototherapy and exchange transfusions. Two weeks post-partum the hyperbilirubinaemia persisted and hepatosplenomegaly became evident. The infant's condition worsened. Weight gain was poor and spontaneous fractures occurred. Liver function tests deteriorated, blood ammonia levels rose constantly. Prothrombin time was prolonged and low fibrinogen levels were found. A t 4 months of age the infant died of progressive lung insufficiency due to CMV pneumonitis complicated by bacterial infection. The other infant had no problems and was transferred to the same neonatal centre at 2 weeks of age for additional investigations. A t admission no physical signs of CMV infection were found even though liver function tests were slightly elevated, Specific antibodies for
262 CMV were present and CMV was detected in the infant's urine. In the following months hepatosplenomegaly developed and progressed to 5 cm below the costal margin at 5 months of age. At this age no other abnormalities were found and liver function tests returned to normal values. The child showed a normal growth pattern. CMV status of the mother 2.5 months after parturition showed positve IgG-antibodies and negative IgM-antibodies. Data concerning her CMV status before or during pregnancy could not be retrieved.
this suggestion are the low transmission rate, the low percentage of severely affected infants, CMV potentially affecting the fetus in all trimesters of pregnancy, and the limited value of serological examinations [2, 6, 7, 10]. Nevertheless, in cases of intra-uterine growth retardation antibody screening for CMV may be of value and may influence obstetric management. As long as no medical treatment of CMV infection is available, treatment of both the affected pregnant mother and the fetus remains symptomatic.
Discussion
References
This case offered the opportunity to observe the different course of CMV infection in a premature twins. Data in literature on CMV infection in twin pregnancies are limited [8]. Congenital CMV infection was demonstrated in these two infants by isolation of the virus from urine and detection of IgM antibodies in cord serum, which may be considered as evidence for an intra-uterine infection that is presumed to be the result of transplacental virus transfer [5]. It has to be assumed that both twins were exposed to CMV at the same time and to the same extent. In this case it is clear that the small fetus was much more affected by the intra-uterine acquired CMV infection than the large fetus. Although it could not be determined in which trimester the CMV infection occurred it is supposed that this took place just before parturition because of the lack of symptoms at birth. The combination of growth retardation and intra-utefine viral infection causing an impaired immunological response might expIain the difference in fetal involvement. Theoretically the donor semen used for artificial insemination could have been the source of this CMV infection. Unfortunately the donor was not available for screening for CMV infection. Thrombocytopenia, coagulation abnormalities and haemolysis, as seen in CMV infection, might increase the risk for complications such as intra-ventricular haemmorrhage. To prevent birth of severely affected infants, antenatal screening for CMV antibodies is suggested [3]. Arguments against
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