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Cognitive and fine motor deficits in a pediatric sickle cell disease cohort of mixed ethnic origin

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Abstract

Cerebrovascular disease is an important feature of pediatric sickle cell disease (SCD) and may lead to cognitive and motor impairment. Our cross-sectional study examined the incidence and severity of these impairments in a pediatric cohort without clinical cerebrovascular events from Berlin of mixed ethnic origin. Thirty-two SCD patients (mean age 11.14 years, range 7.0–17.25 years; males 14) were evaluated for full-scale intelligence (IQ) (German version WISC-III), fine motor function (digital writing tablet), and executive function (planning, attention, working memory, and visual-spatial abilities) with the Amsterdam Neuropsychological Tasks (ANT) program and the Tower of London (ToL). Data on clinical risk factors were retrieved from medical records. Full-scale IQ of patients was preserved, whereas performance IQ was significantly reduced (91.19 (SD 12.17) d = 0.7, p = 0.007). SCD patients scored significantly lower than healthy peers when tested for executive and fine motor functions, e.g., planning time in the ToL (6.73 s (SD 3.21) vs. 5.9 s in healthy peers (SD 2.33), d = 0.5, p = <0.001) and frequency on the writing tablet (mean z score −0.79, d = 0.7, p < 0.001). No clinical risk factors were significantly associated with incidence and severity of cognitive and motor deficits. Despite the preservation of full-scale IQ, our SCD cohort of mixed origin exhibited inferior executive abilities and reduced fine motor skills. Our study is limited by the small size of our cohort as well as the lack for control of sociodemographic and socioeconomic factors modulating higher functions but highlights the need for early screening, prevention, and specific interventions for these deficits.

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Acknowledgments

We gratefully thank “KINDerLEBENe.V.” for supporting Elisabeth Koustenis and the “Kind-Philipp-Foundation” for supporting Stefan Mark Rueckriegel. We are indebted to Raffaella Colombatti for many helpful discussions and critical reading of the manuscript. We also thank Hermann Baqué for support when performing the cognitive tests.

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    1. Klinik für Pädiatrie m.S. Onkologie/Hämatologie/KMT, Charité – Universitätsmedizin Berlin, Augustenburger Platz 1, 13353, Berlin, Germany

      Luise Burkhardt, Stephan Lobitz, Elisabeth Koustenis, Stefan Mark Rueckriegel & Pablo Hernáiz Driever

    2. Present address: Universitätsklinikum S-H. Medizinische Klinik II und Poliklinik m.S. Onkologie/Hämatologie, Arnold-Heller-Straße 3, 24105, Kiel, Germany

      Luise Burkhardt

    3. Pädiatrie 5- Klinik für Pädiatrie m. S. Onkologie, Hämatologie und Immunologie, Olgahospital Stuttgart, Kriegsbergstr. 62, 70174, Stuttgart, Germany

      Elisabeth Koustenis

    4. Department of Neurosurgery, University Hospital Würzburg, Würzburg, Germany

      Stefan Mark Rueckriegel

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    Luise Burkhardt and Stephan Lobitz contributed equally to this work.

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    Burkhardt, L., Lobitz, S., Koustenis, E. et al. Cognitive and fine motor deficits in a pediatric sickle cell disease cohort of mixed ethnic origin. Ann Hematol 96, 199–213 (2017). https://doi.org/10.1007/s00277-016-2861-1

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