David O Kamson1, Csaba Juhász2, Joseph Shin3, Michael E Behen1, William C Guy3, Harry T Chugani2, Jeong-Won Jeong4. 1. Carman and Ann Adams Department of Pediatrics, Wayne State University School of Medicine, Detroit, Michigan; Translational Imaging Laboratory, Children's Hospital of Michigan, Detroit, Michigan. 2. Carman and Ann Adams Department of Pediatrics, Wayne State University School of Medicine, Detroit, Michigan; Department of Neurology, Wayne State University School of Medicine, Detroit, Michigan; Translational Imaging Laboratory, Children's Hospital of Michigan, Detroit, Michigan. 3. Translational Imaging Laboratory, Children's Hospital of Michigan, Detroit, Michigan. 4. Carman and Ann Adams Department of Pediatrics, Wayne State University School of Medicine, Detroit, Michigan; Department of Neurology, Wayne State University School of Medicine, Detroit, Michigan; Translational Imaging Laboratory, Children's Hospital of Michigan, Detroit, Michigan. Electronic address: jeongwon@pet.wayne.edu.
Abstract
BACKGROUND: Reorganization of the corticospinal tract after early damage can limit motor deficit. In this study, we explored patterns of structural corticospinal tract reorganization in children with Sturge-Weber syndrome. METHODS: Five children (age 1.5-7 years) with motor deficit resulting from unilateral Sturge-Weber syndrome were studied prospectively and longitudinally (1-2 years follow-up). Corticospinal tract segments belonging to hand and leg movements were separated and their volume was measured by diffusion tensor imaging tractography using a recently validated method. Corticospinal tract segmental volumes were normalized and compared between the Sturge-Weber syndrome children and age-matched healthy controls. Volume changes during follow-up were also compared with clinical motor symptoms. RESULTS: In the Sturge-Weber syndrome children, hand-related (but not leg-related) corticospinal tract volumes were consistently decreased in the affected cerebral hemisphere at baseline. At follow-up, two distinct patterns of hand corticospinal tract volume changes emerged. (1) Two children with extensive frontal lobe damage showed a corticospinal tract volume decrease in the lesional hemisphere and a concomitant increase in the nonlesional (contralateral) hemisphere. These children developed good hand grasp but no fine motor skills. (2) The three other children, with relative sparing of the frontal lobe, showed an interval increase of the normalized hand corticospinal tract volume in the affected hemisphere; these children showed no gross motor deficit at follow-up. CONCLUSIONS: Diffusion tensor imaging tractography can detect differential abnormalities in the hand corticospinal tract segment both ipsi- and contralateral to the lesion. Interval increase in the corticospinal tract hand segment suggests structural reorganization, whose pattern may determine clinical motor outcome and could guide strategies for early motor intervention.
BACKGROUND: Reorganization of the corticospinal tract after early damage can limit motor deficit. In this study, we explored patterns of structural corticospinal tract reorganization in children with Sturge-Weber syndrome. METHODS: Five children (age 1.5-7 years) with motor deficit resulting from unilateral Sturge-Weber syndrome were studied prospectively and longitudinally (1-2 years follow-up). Corticospinal tract segments belonging to hand and leg movements were separated and their volume was measured by diffusion tensor imaging tractography using a recently validated method. Corticospinal tract segmental volumes were normalized and compared between the Sturge-Weber syndromechildren and age-matched healthy controls. Volume changes during follow-up were also compared with clinical motor symptoms. RESULTS: In the Sturge-Weber syndromechildren, hand-related (but not leg-related) corticospinal tract volumes were consistently decreased in the affected cerebral hemisphere at baseline. At follow-up, two distinct patterns of hand corticospinal tract volume changes emerged. (1) Two children with extensive frontal lobe damage showed a corticospinal tract volume decrease in the lesional hemisphere and a concomitant increase in the nonlesional (contralateral) hemisphere. These children developed good hand grasp but no fine motor skills. (2) The three other children, with relative sparing of the frontal lobe, showed an interval increase of the normalized hand corticospinal tract volume in the affected hemisphere; these children showed no gross motor deficit at follow-up. CONCLUSIONS: Diffusion tensor imaging tractography can detect differential abnormalities in the hand corticospinal tract segment both ipsi- and contralateral to the lesion. Interval increase in the corticospinal tract hand segment suggests structural reorganization, whose pattern may determine clinical motor outcome and could guide strategies for early motor intervention.
Authors: Judith D Schaechter; Zachary P Fricker; Katherine L Perdue; Karl G Helmer; Mark G Vangel; Douglas N Greve; Nikos Makris Journal: Hum Brain Mapp Date: 2009-11 Impact factor: 5.038
Authors: Alejandro J De la Torre; Aimee F Luat; Csaba Juhász; Mai Lan Ho; Davis P Argersinger; Kara M Cavuoto; Mabel Enriquez-Algeciras; Stephanie Tikkanen; Paula North; Craig N Burkhart; Harry T Chugani; Karen L Ball; Anna Lecticia Pinto; Jeffrey A Loeb Journal: Pediatr Neurol Date: 2018-04-18 Impact factor: 3.372