ObjectiveTo evaluate clinical, audiological and neuroimaging findings in a cohort of infants diagnosed with congenital cytomegalovirus (cCMV) infection after failure at newborn hearing screening.... Show moreObjectiveTo evaluate clinical, audiological and neuroimaging findings in a cohort of infants diagnosed with congenital cytomegalovirus (cCMV) infection after failure at newborn hearing screening. MethodsA prospective observational study in the Netherlands, using the existing newborn hearing screening infrastructure for well babies. Between July 2012 and November 2016, cytomegalovirus (CMV) PCR testing of neonatally obtained dried blood spots (DBS) was offered to all infants who failed newborn hearing screening. Clinical, neuroimaging and audiological data were collected. ResultsDBS of 1374 infants were successfully tested and 59 were positive for CMV (4.3%). Data of 54 infants were retrieved. Three were small for gestational age and six had microcephaly. Forty-eight (89%) had sensorineural hearing loss (SNHL), of whom half had unilateral SNHL. In both unilaterally and bilaterally affected children, the majority of the impaired ears had severe or profound hearing loss. Neuroimaging abnormalities were found in 40 of 48 (83%) children who had evaluable cranial ultrasound and/or cerebral MRI. The abnormalities were mild in 34, moderate in 3 and severe in 3 infants. The degree of SNHL and the severity of neuroimaging abnormalities were found to be correlated (p=0.002). ConclusionsThe yield of targeted cCMV screening following newborn hearing screening failure was eight times higher than the estimated national birth prevalence of cCMV. The majority of this cohort of infants with clinically unsuspected cCMV disease had confirmed SNHL, neuroimaging abnormalities and lower than average birth weights and head circumferences. Newborns who fail newborn hearing screening should be tested for CMV to ensure appropriate clinical, neurodevelopmental and audiological follow-up. Show less
Objectives: To compare the quality of life (QoL) of children with hearing loss (HL) and children with normal hearing (NH) and to examine how the QoL of children with HL changes over time,... Show moreObjectives: To compare the quality of life (QoL) of children with hearing loss (HL) and children with normal hearing (NH) and to examine how the QoL of children with HL changes over time, considering language skills, type of hearing device, degree of HL, and type of education.Methods and materials: This longitudinal study included 62 children with HL and their parents. Developmental outcome data were collected at two time points, when the mean ages of the children were 4 and 11 years. The Pediatric Quality of Life (PedsQL (TM)) questionnaire, which includes assessments of Physical, Emotional, Social, and School functioning, was completed by parents at both time points and by the children with HL at the second time point. Receptive and expressive language skills at 4 years were assessed by the Reynell Developmental Language Scale. Results were compared with a Dutch normative sample.Results: The QoL of children with HL was similar to that of children with NH at both time points on two of the four QoL scales, Emotional and Physical functioning. On the other two scales, Social and School functioning, children with HL who attended special education and children who switched to mainstream education showed lower scores than children with HL who were consistently in mainstream education and lower scores than children with NH. The School QoL of children with HL decreased over time, as did the School QoL of children with NH. Social QoL of children with cochlear implants decreased over time, but this was not the case in children with hearing aids. Language skills and the degree of HL did not clinically improve the QoL over time of preschool children with HL.Conclusions: The QoL of children with HL in mainstream education and the Physical and Emotional QoL of all children with HL were satisfactory. It is essential to develop specific guidance regarding school activities for children with HL in special education and for children with HL who switch to mainstream education in order to increase their social QoL. Show less
Objectives: To compare the quality of life (QoL) of children with hearing loss (HL) and children with normal hearing (NH) and to examine how the QoL of children with HL changes over time,... Show moreObjectives: To compare the quality of life (QoL) of children with hearing loss (HL) and children with normal hearing (NH) and to examine how the QoL of children with HL changes over time, considering language skills, type of hearing device, degree of HL, and type of education.Methods and materials: This longitudinal study included 62 children with HL and their parents. Developmental outcome data were collected at two time points, when the mean ages of the children were 4 and 11 years. The Pediatric Quality of Life (PedsQL (TM)) questionnaire, which includes assessments of Physical, Emotional, Social, and School functioning, was completed by parents at both time points and by the children with HL at the second time point. Receptive and expressive language skills at 4 years were assessed by the Reynell Developmental Language Scale. Results were compared with a Dutch normative sample.Results: The QoL of children with HL was similar to that of children with NH at both time points on two of the four QoL scales, Emotional and Physical functioning. On the other two scales, Social and School functioning, children with HL who attended special education and children who switched to mainstream education showed lower scores than children with HL who were consistently in mainstream education and lower scores than children with NH. The School QoL of children with HL decreased over time, as did the School QoL of children with NH. Social QoL of children with cochlear implants decreased over time, but this was not the case in children with hearing aids. Language skills and the degree of HL did not clinically improve the QoL over time of preschool children with HL.Conclusions: The QoL of children with HL in mainstream education and the Physical and Emotional QoL of all children with HL were satisfactory. It is essential to develop specific guidance regarding school activities for children with HL in special education and for children with HL who switch to mainstream education in order to increase their social QoL. Show less
Korndewal, M.J.; Weltevrede, M.; Akker-van Marle, M.E. van den; Oudesluys-Murphy, A.M.; Melker, H.E. de; Vossen, A.C.T.M. 2018
Background Delayed language development without an obvious cause is considered an isolated developmental disorder and is called specific language impairment (SLI). SLI is probably the most... Show moreBackground Delayed language development without an obvious cause is considered an isolated developmental disorder and is called specific language impairment (SLI). SLI is probably the most prevalent developmental disorder in childhood with a generally cited prevalence of 7 %. This study aimed to investigate whether SLI is always an isolated disorder or if children with SLI also have delayed motor development. Methods We used data of an earlier study with a prospective nested case-control design in which developmental data were collected from child healthcare files. Cases were children (4-11 years) with diagnosed SLI. They were matched by sex and date of birth with control children attending mainstream education. Data of both groups on seven gross and six fine motor milestones which had been registered in the Dutch Developmental Instrument between the ages of 15- 36 months were retrieved from child healthcare files. McNemar tests were performed to test for differences in reaching motor milestones at the age norm between the case and control group. Results Data from 253 children in each group were available. A significant difference was found between both groups in the proportion failing to reach three of the seven investigated gross motor milestones at the age norm (p < 0.05). The proportion of children not reaching the motor milestone at the age norm was significantly higher for five of the six fine motor milestones in children with SLI compared with control children (p < 0.05). Conclusions More children with SLI are late in reaching motor milestones than children without SLI. This means that it is debatable whether SLI can be regarded as a "specific" impairment which is not associated with other developmental problems. A broader developmental assessment is therefore indicated when diagnosing SLI. Show less
Congenital cytomegalovirus infection (cCMV) may lead to symptoms at birth and long-term consequences. We present a nationwide, retrospective cohort study on the outcome of cCMV up to age 6 years.... Show moreCongenital cytomegalovirus infection (cCMV) may lead to symptoms at birth and long-term consequences. We present a nationwide, retrospective cohort study on the outcome of cCMV up to age 6 years. For this study we identified cCMV, using polymerase chain reaction, by analysing dried blood spots, which are taken shortly after birth for neonatal screening. The group of children with cCMV were compared to a group of children who were cCMV negative at birth. Data were collected about their health and development up to age 6 years. Parents of 73 693 children were invited to participate, and 32 486 (44.1%) gave informed consent for testing of their child's dried blood spot for CMV. Of the 31 484 dried blood spots tested, 156 (0.5%) were positive for cCMV. Of these, four (2.6%) children had been diagnosed with cCMV prior to this study. This unique retrospective nationwide study permits the estimation of long-term sequelae of cCMV up to the age of 6 years. The birth prevalence of cCMV in this study was 0.5%, which is in line with prior estimates. Most (97.4%) children with cCMV had not been diagnosed earlier, indicating under-diagnosis of cCMV. Show less
