Hemophilia Growth and Development Study: Relationships Between Neuropsychological, Neurological, and MRI Findings at Baseline

Sirois, Patricia A.; Usner, Dale W; Hill, Stéphanie; Mitchell, Wendy G.; Bale, James F.; Loveland, Katherine A.; Stehbens, James A.; Donfield, Sharyne; Maeder, Margaret A.; Amodei, Nancy; Contant, Charles F.; Nelson, Marvin D.; Willis, John

doi:10.1093/jpepsy/23.1.45

Cited by 19 publications

(40 citation statements)

References 17 publications

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“…Span percent correct and latency to correct were associated with the presence of a premorbid history of intracerebral hemorrhage, but were not sensitive to HIV status. This result parallels Sirois et al (1998), also reporting on the HGDS sample, who found that a documented history of head trauma was associated with lowered neuropsychological performance in both HIV1 and HIV2 groups. History of head trauma was relatively common in both HIV1 and HIV2 groups in both Sirois et al (1998) and the present study.…”

Section: Discussionsupporting

confidence: 88%

“…However, follow-up of HGDS participants over a period of 5 years revealed significant declines in neuropsychological performance that directly related to declines in immune functioning (Loveland et al, in press). Although neuropsychological deficits were not associated with HIV infection or immune suppression at baseline, lowered neuropsychological performance was found in HGDS participants with a history of head trauma and coordination and0or gait abnormalities that probably reflect hemophilia-related morbidity (Sirois et al, 1998). The results of both the Loveland et al (1994) and Sirois et al (1998) studies were interpreted as showing that adaptive behavioral, academic, and cognitive difficulties in the baseline HGDS sample reflect the effects of hemophilia as a chronic illness rather than the effects of HIV infection.…”

Section: The Hemophilia Growth and Development Studymentioning

confidence: 93%

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Attention in HIV-infected children: Results from the Hemophilia Growth and Development Study

Watkins

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Usner

et al. 2000

J Int Neuropsychol Soc

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Attentional functioning was examined in three groups of 7-to 19-year-old male participants with hemophilia:(1) HIV seronegative controls (HIV2, N 5 66), (2) HIV seropositive participants with CD41 lymphocyte counts greater than or equal to 200 (HIV1 CD41 Ն200, N 5 79), and (3) severely immune suppressed HIV seropositive participants (HIV1 CD41 ,200, N 5 28). Two measures sensitive to attention deficits were used: the Continuous Performance Test (CPT) and the Span of Apprehension (Span). On the CPT, there was a decrement in attention in both HIV1 groups, as indexed by an increase in false alarm rate from Block 1 to Block 3, that was not present in the HIV2 group. The longer the HIV1 children were required to sustain attention to the CPT, the more they responded to the incorrect stimulus. This effect decreased as age increased. Span percent correct and latency to correct were associated with the presence of a premorbid history of intracerebral hemorrhage, but were not sensitive to HIV status or degree of immune suppression in the HIV1 children, suggesting morbidity related to hemophilia. The remaining CPT and Span variables-hit rate, sensitivity, latency, percent correct, and latency to correctshowed the expected associations with age, but none showed conclusive associations with HIV status or immune suppression in the HIV1 participants. (JINS, 2000, 6, 443-454.)

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Section: Discussionsupporting

confidence: 88%

Section: The Hemophilia Growth and Development Studymentioning

confidence: 93%

Attention in HIV-infected children: Results from the Hemophilia Growth and Development Study

Watkins

Cool

Usner

et al. 2000

J Int Neuropsychol Soc

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Section: Vs 413)mentioning

confidence: 99%

“…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. 11,12 Another HGDS report showed associations between prolonged hospitalization and lower achievement test scores in reading and spelling (Wong WY et al, unpublished data).To extend observations made in the HGDS, the Academic Achievement in Children with Hemophilia study group was formed. Investigators from 18 comprehensive US hemophilia treatment centers collaborated in the collection of social, demographic, hemophilia-related, and psychological data from a population-based cohort of young school-aged children with severe hemophilia in an effort to further define relationships between hemophilia and academic performance.…”

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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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“…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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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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Hemophilia Growth and Development Study: Relationships Between Neuropsychological, Neurological, and MRI Findings at Baseline

Abstract: Hemophilia-related morbidity has a subtle adverse influence on cognitive performance. HIV infection was not associated with neuropsychological dysfunction in this group even when MRI abnormalities were present.

Cited by 19 publications

References 17 publications

Attention in HIV-infected children: Results from the Hemophilia Growth and Development Study

Attention in HIV-infected children: Results from the Hemophilia Growth and Development 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

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