Hemophilia Growth and Development Study: Baseline Neurodevelopmental Findings

Loveland, Katherine A.; Stehbens, James A.; Contant, Charles F.; Bordeaux, Janice D.; Sirois, Patricia A.; Bell, Terece S.; Hill, Stéphanie; Scott, Anthony A.; Bowman, Mary Jean; Schiller, Marilyn; Watkins, John M.; Olson, Roberta A.; Moylan, Patricia M.; Cool, Valerie A.; Belden, Brain

doi:10.1093/jpepsy/19.2.223

Cited by 46 publications

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“…Higher school absenteeism rates compared with their nonaffected classmates 6,9 and lower academic performance relative to potential have also been documented. 10 The primary occupation of young children is preparation for and participation at school. Factors that interfere with completion of these important tasks can result in the inability of the child to fulfill his or her potential during school and later in life.…”

Section: Discussionmentioning

confidence: 99%

“…5 Several reports in the literature document higher rates of school absenteeism in children with hemophilia than in others 6 -9 and discrepancies between children's potential and achievement as they progress through the upper grades. 8 Studying children enrolled in the multicenter Hemophilia Growth and Development Study (HGDS), Loveland et al 10 reported lower achievement than expected based on IQ at the baseline examination. Analyses of baseline and longitudinally collected HGDS data demonstrated associations between the functional severity of hemophilia, measured by abnormalities in coordination and gait, and academic performance.…”

Section: Vs 413)mentioning

confidence: 99%

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Defining the Impact of Hemophilia: The Academic Achievement in Children With Hemophilia Study

et al. 2001

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ABSTRACT. Objectives. We characterized a population-based cohort of school-aged children with severe hemophilia with respect to type of treatment, on-demand versus prophylaxis, and frequency of bleeding episodes in the year before enrollment. We also investigated the association between hemophilia-related morbidity, measured by number of bleeding episodes in the year before enrollment, and academic performance after adjustment for other factors known to have an effect on achievement. Finally, we explored the mechanisms for the association between bleeding episodes and academic achievement.Study Design. This study was a multicenter investigation of boys 6 to 12 years old with severe factor VIII deficiency (clotting factor level <2%) receiving care in US hemophilia treatment centers. Children with a history of inhibitor, severe developmental disorder, significant psychiatric disorder, or insufficient fluency in English were excluded from the study. On-demand treatment was defined as administration of clotting factor on the occurrence of a bleeding episode. Prophylactic therapy was defined as a course of regular infusions for >2 months with a goal of preventing bleeding episodes. Academic achievement was measured by the Wechsler Individual Achievement Test. Quality of life was measured by the Child Health Questionnaire. Of particular interest was the Physical Summary (PhS) measure of the Child Health Questionnaire. The type of information captured by the PhS includes limitations in physical activity, limitations in the kind or amount of schoolwork or social activities the child engaged in, and presence of pain or discomfort.Results. One hundred thirty-one children were enrolled, a median center recruitment rate of 77%. The mean age of the participants was 9.6 years, and approximately half of the participants had completed less than the fourth grade at the time of enrollment. Sixty-two percent of the children were on prophylaxis at enrollment, and 9% had previously been on prophylaxis but were currently on on-demand therapy. Two groups were defined: ever treated with prophylaxis and never treated with prophylaxis. For those ever treated, treatment duration ranged from 2.7 months to 7.7 years, with one half of the children treated with prophylaxis for >40% of their lifetimes; 29% had always been on on-demand therapy. Children in both treatment groups were similar with respect to age, clotting factor level, parents' education, and IQ. The median number of bleeding episodes experienced in the year before enrollment for the cohort as a whole was 12. The median number of bleeding episodes in children on prophylaxis at enrollment was significantly lower than in children on on-demand therapy (6 vs 25.5).The mean achievement scores were within the average range of academic performance: reading, 100.4; mathematics, 101.6; language, 108.1; writing, 95.4; and total achievement, 102.5. When children were categorized as above or below the study group median by number of bleeding episodes, those who had a low number of bleeding e...

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Section: Discussionmentioning

confidence: 99%

Section: Vs 413)mentioning

confidence: 99%

Defining the Impact of Hemophilia: The Academic Achievement in Children With Hemophilia Study

et al. 2001

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“…1,27,29 -33 However, Cohen et al 34 did not find significant differences between HIV-infected and noninfected children in global intellectual ability, even 4 1 ⁄2 to 8 1 ⁄2 years after onset, and others [35][36][37] have noted that neuropsychological profiles in HIV-infected and noninfected hemophiliacs did not differ. Thus, infants and children infected with HIV infection have, sometimes, but not always, been found to have deficits in global intellectual functioning.…”

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confidence: 95%

Predicting HIV Disease Progression in Children Using Measures of Neuropsychological and Neurological Functioning

et al. 2000

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ABSTRACT. Background. Neuropsychological testing and 2 measures of neurological status, cortical atrophy, and motor dysfunction were assessed for their usefulness in predicting human immunodeficiency virus (HIV) disease progression in infants, children, and adolescents who participated in Pediatric AIDS Clinical Trials Group Protocol 152 (PACTG 152).Methods. A cohort of 722 antiretroviral therapy-naive children with symptomatic HIV infection were assessed at study entry and at later intervals. Assessments included neurodevelopmental testing, neuroradiologic imaging, and neurological examination of motor function. CD4 cell count and plasma RNA viral load also were measured.Results. Children with the lowest neuropsychological functioning (IQ < 70) at baseline had the highest risk for later HIV disease progression (56%), compared with those with borderline/low (IQ ‫؍‬ 70 -89) functioning (26%), or with average or above (IQ > 90) functioning (18%). This was also true of week 48 neuropsychological functioning. Motor dysfunction (especially reduced muscle mass) at entry also predicted disease progression. Furthermore, motor dysfunction and week 48 neuropsychological functioning provided predictive information beyond that obtainable from surrogate markers of HIV disease status (eg, CD4 count, HIV RNA level). Children with cortical atrophy also were at higher risk for later disease progression, but when CD4 count and RNA viral load were known, cortical atrophy information provided no additional predictive information.Conclusions. Measures of neuropsychological and motor function status provide unique information regarding pediatric HIV disease progression. As such, these findings have important implications for predicting long-term outcomes (eg, longevity) in pediatric patients. Pediatrics 2000;106(6). URL: http://www. pediatrics.org/cgi/content/full/106/6/e76; human immunodeficiency virus, children, neuropsychology, cognitive, development, cortical atrophy, motor function.

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“…Similarly, abnormalities on neuroimaging were limited to the same group [Mitchell et al, 1993]. Seropositive subjects did not differ on most aspects of neuropsychological testing from seronegative subjects [Loveland et al, 1994]. Longitudinal followup of the group revealed that behavioral changes and diffuse muscle atrophy were the only significant neurological findings attributable to HIV, and only very late in the course, usually in the six months preceding death [Mitchell et al, 1997].…”

Section: Cognitive Functioning Of Older Children With Hiv Infection Amentioning

confidence: 97%

Neurological and developmental effects of HIV and AIDS in children and adolescents

Mitchell

2001

Ment. Retard. Dev. Disabil. Res. Rev.

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HIV-related encephalopathy is an important problem in vertically infected children with HIV. Infected infants may manifest early, catastrophic encephalopathy, with loss of brain growth, motor abnormalities, and cognitive dysfunction. Even without evidence of AIDS, infected infants score lower than serorevertors on developmental measures, particularly language acquisition. Children with perinatal or later transfusion-related infection generally are roughly comparable developmentally to their peers until late in their course. Symptoms similar to adult AIDS dementia complex are occasionally seen in adolescents with advanced AIDS, including dementia, bradykinesia, and spasticity. Opportunistic CNS infections such as toxoplasmosis and progressive multifocal leukoencephalopathy are less common in children and adolescents than in adults. Increasing evidence suggests that aggressive antiretroviral treatment may halt or even reverse encephalopathy. Neuroimaging changes may precede or follow clinical manifestations, and include early lenticulostriate vessel echogenicity on cranial ultrasound, calcifying microangiopathy on CT scan, and/or white matter lesions and central atrophy on MRI. Differential diagnosis of neurological dysfunction in an HIV-infected infant includes the effects of maternal substance abuse, other CNS congenital infections, and other causes of early static encephalopathy. Initial entry of HIV into the nervous system occurs very early in infection. The risk of clinical HIV encephalopathy increases with very early age of infection and with high viral loads. Virus is found in microglia and brain derived macrophages, not neurons. The neuronal effect of HIV is probably indirect, with various cytokines implicated. Apoptosis is the presumed mechanism of damage to neurons by HIV.

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Hemophilia Growth and Development Study: Baseline Neurodevelopmental Findings

Cited by 46 publications

References 0 publications

Defining the Impact of Hemophilia: The Academic Achievement in Children With Hemophilia Study

Defining the Impact of Hemophilia: The Academic Achievement in Children With Hemophilia Study

Predicting HIV Disease Progression in Children Using Measures of Neuropsychological and Neurological Functioning

Neurological and developmental effects of HIV and AIDS in children and adolescents

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