Daily Quantity and Kinematic Characteristics of Leg Movement in a Child With SMA (2 Copies SMN2)

被引:2
作者
Mcintyre, Melissa [1 ,5 ]
Dunn, Lauren [2 ]
David, Jed [2 ]
Devine, Ceri
Smith, Beth A. [3 ,4 ,6 ]
机构
[1] Univ Utah, Dept Pediat, Salt Lake City, UT USA
[2] Childrens Hosp Los Angeles, Dept Rehabil Serv, Los Angeles, CA USA
[3] Childrens Hosp Los Angeles, Saban Res Inst, Dev Neurosci & Neurogenet Program, Div Dev Behav Pediat, Los Angeles, CA USA
[4] Univ Southern Calif, Keck Sch Med, Dept Pediat, Los Angeles, CA USA
[5] Univ Utah, Dept Pediat, 15 North 2030 East,EIGH 2250A, Salt Lake City, UT 84112 USA
[6] Childrens Hosp Los Angeles, Saban Res Inst, Dev Neurosci & Neurogenet Program, Div Dev Behav Pediat, 4650 Sunset Blvd,MS 183, Los Angeles, CA 90027 USA
关键词
Spinal Muscular Atrophy; wearable sensors; SPINAL MUSCULAR-ATROPHY; SHAM CONTROL; DIAGNOSIS; NUSINERSEN;
D O I
10.1097/PEP.0000000000001053
中图分类号
R72 [儿科学];
学科分类号
100202 ;
摘要
Purpose: This case report describes daily leg movement quantity and kinematic characteristics of a child with spinal muscular atrophy (SMA) with 2 copies of SMN2, who was symptomatic at the time of treatment with disease-modifying therapies.Key Points: Compared with infants with typical development, this child had differing values for leg movement quantity, duration, average acceleration, and peak acceleration measured across full days in the natural environment by wearable sensors. In addition, movement quantity and clinician-rated outcomes increased with age.Conclusions: Wearable sensors recorded movement quantity and kinematic characteristics in a treated infant with SMA (2 copies SMN2). These movement parameters were consistently different compared anecdotally with published data from infants with typical development, demonstrating their potential to add unique and complementary information to the assessment of motor function in SMA.Recommendations: Larger longitudinal studies are needed to determine the utility of wearable sensors as an assessment tool and an early predictor of motor outcomes in children with SMA.
引用
收藏
页码:486 / 492
页数:7
相关论文
共 28 条
  • [1] Identification of Developmental Delay in Infants Using Wearable Sensors: Full-Day Leg Movement Statistical Feature Analysis
    Abrishami, Mohammad Saeed
    Nocera, Luciano
    Mert, Melissa
    Trujillo-Priego, Ivan A.
    Purushotham, Sanjay
    Shahabi, Cyrus
    Smith, Beth A.
    [J]. IEEE JOURNAL OF TRANSLATIONAL ENGINEERING IN HEALTH AND MEDICINE, 2019, 7
  • [2] ACTIVE (Ability Captured Through Interactive Video Evaluation) workspace volume video game to quantify meaningful change in spinal muscular atrophy
    Alfano, Lindsay N.
    Miller, Natalie F.
    Iammarino, Megan A.
    Moore Clingenpeel, Melissa
    Lowes, Suzanne L.
    Dugan, Margaret E.
    Kissel, John T.
    Al Zaidy, Samiah
    Tsao, Chang-Yong
    Lowes, Linda P.
    [J]. DEVELOPMENTAL MEDICINE AND CHILD NEUROLOGY, 2020, 62 (03) : 303 - +
  • [3] Natural history of Type 2 and 3 spinal muscular atrophy: 2-year NatHis-SMA study
    Annoussamy, Melanie
    Seferian, Andreea M.
    Daron, Aurore
    Pereon, Yann
    Cances, Claude
    Vuillerot, Carole
    De Waele, Liesbeth
    Laugel, Vincent
    Schara, Ulrike
    Gidaro, Teresa
    Lilien, Charlotte
    Hogrel, Jean-Yves
    Carlier, Pierre
    Fournier, Emmanuel
    Lowes, Linda
    Gorni, Ksenija
    Ly-Le Moal, Myriam
    Hellbach, Nicole
    Seabrook, Timothy
    Czech, Christian
    Hermosilla, Ricardo
    Servais, Laurent
    [J]. ANNALS OF CLINICAL AND TRANSLATIONAL NEUROLOGY, 2021, 8 (02): : 359 - 373
  • [4] Spinal Muscular Atrophy Treatments, Newborn Screening, and the Creation of a Neurogenetics Urgency
    Butterfield, Russell J.
    [J]. SEMINARS IN PEDIATRIC NEUROLOGY, 2021, 38
  • [5] Correlation between SMA type and SMN2 copy number revisited: An analysis of 625 unrelated Spanish patients and a compilation of 2834 reported cases
    Calucho, Maite
    Bernal, Sara
    Alias, Laura
    March, Francesca
    Vencesla, Adoracion
    Rodriguez-Alvarez, Francisco J.
    Aller, Elena
    Fernandez, Raquel M.
    Borrego, Salud
    Milian, Jose M.
    Hernandez-Chico, Concepcion
    Cusco, Ivon
    Fuentes-Prior, Pablo
    Tizzano, Eduardo F.
    [J]. NEUROMUSCULAR DISORDERS, 2018, 28 (03) : 208 - 215
  • [6] Nusinersen initiated in infants during the presymptomatic stage of spinal muscular atrophy: Interim efficacy and safety results from the Phase 2 NURTURE study
    De Vivo, Darryl C.
    Bertini, Enrico
    Swoboda, Kathryn J.
    Hwu, Wuh-Liang
    Crawford, Thomas O.
    Finkel, Richard S.
    Kirschner, Janbernd
    Kuntz, Nancy L.
    Parsons, Julie A.
    Ryan, Monique M.
    Butterfield, Russell J.
    Topaloglu, Haluk
    Ben-Omran, Tawfeg
    Sansone, Valeria A.
    Jong, Yuh-Jyh
    Shu, Francy
    Staropoli, John F.
    Kerr, Douglas
    Sandrock, Alfred W.
    Stebbins, Christopher
    Petrillo, Marco
    Braley, Gabriel
    Johnson, Kristina
    Foster, Richard
    Gheuens, Sarah
    Bhan, Ishir
    Reyna, Sandra P.
    Fradette, Stephanie
    Farwell, Wildon
    [J]. NEUROMUSCULAR DISORDERS, 2019, 29 (11) : 842 - 856
  • [7] Deng Weiyang, 2018, J Mot Learn Dev, V6, P101, DOI 10.1123/jmld.2016-0056
  • [8] Nusinersen versus Sham Control in Infantile-Onset Spinal Muscular Atrophy
    Finkel, R. S.
    Mercuri, E.
    Darras, B. T.
    Connolly, A. M.
    Kuntz, N. L.
    Kirschner, J.
    Chiriboga, C. A.
    Saito, K.
    Servais, L.
    Tizzano, E.
    Topaloglu, H.
    Tulinius, M.
    Montes, J.
    Glanzman, A. M.
    Bishop, K.
    Zhong, Z. J.
    Gheuens, S.
    Bennett, C. F.
    Schneider, E.
    Farwell, W.
    De Vivo, D. C.
    [J]. NEW ENGLAND JOURNAL OF MEDICINE, 2017, 377 (18) : 1723 - 1732
  • [9] Pre-symptomatic spinal muscular atrophy: a proposed nosology COMMENT
    Finkel, Richard S.
    Benatar, Michael
    [J]. BRAIN, 2022, 145 (07) : 2247 - 2249
  • [10] Toward Predicting Infant Developmental Outcomes From Day-Long Inertial Motion Recordings
    Fitter, Naomi T.
    Funke, Rebecca
    Carlos Pulido, Jose
    Mataric, Maja J.
    Smith, Beth A.
    [J]. IEEE TRANSACTIONS ON NEURAL SYSTEMS AND REHABILITATION ENGINEERING, 2020, 28 (10) : 2305 - 2314