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Three-Dimensional Gait Analysis as a Biomarker for GTP Cyclohydrolase 1-Deficient Dopa-Responsive Dystonia.
Narahara, Sho; Ochi, Nobuhiko; Ito, Yuji; Ito, Tadashi; Narita, Hajime; Noritake, Koji; Kidokoro, Hiroyuki; Natsume, Jun.
Afiliación
  • Narahara S; Department of Pediatrics, Aichi Prefecture Mikawa Aoitori Medical and Rehabilitation Center for Developmental Disabilities, Okazaki, Japan. Electronic address: narahara@mikawa-aoitori.jp.
  • Ochi N; Department of Pediatrics, Aichi Prefecture Mikawa Aoitori Medical and Rehabilitation Center for Developmental Disabilities, Okazaki, Japan.
  • Ito Y; Department of Pediatrics, Aichi Prefecture Mikawa Aoitori Medical and Rehabilitation Center for Developmental Disabilities, Okazaki, Japan; Department of Pediatrics, Nagoya University Graduate School of Medicine, Nagoya, Japan.
  • Ito T; Three-Dimensional Motion Analysis Laboratory, Aichi Prefectural Mikawa Aoitori Medical and Rehabilitation Center for Developmental Disabilities, Okazaki, Japan.
  • Narita H; Department of Pediatrics, Nagoya University Graduate School of Medicine, Nagoya, Japan.
  • Noritake K; Department of Orthopedic Surgery, Aichi Prefecture Mikawa Aoitori Medical and Rehabilitation Center for Developmental Disabilities, Okazaki, Japan.
  • Kidokoro H; Department of Pediatrics, Nagoya University Graduate School of Medicine, Nagoya, Japan.
  • Natsume J; Department of Pediatrics, Nagoya University Graduate School of Medicine, Nagoya, Japan; Department of Developmental Disability Medicine, Nagoya University Graduate School of Medicine, Nagoya, Japan.
Pediatr Neurol ; 154: 66-69, 2024 May.
Article en En | MEDLINE | ID: mdl-38547557
ABSTRACT

BACKGROUND:

GTP-cyclohydrolase 1-deficient dopa-responsive dystonia (GTPCH1-deficient DRD) typically presents in childhood with dystonic posture of the lower extremities, gait impairment, and a significant response to levodopa. We performed three-dimensional gait analysis (3DGA) to quantitatively assess the gait characteristics and changes associated with levodopa treatment in patients with GTPCH1-deficient DRD.

METHODS:

Three levodopa-treated patients with GTPCH1-deficient DRD underwent 3DGA twice, longitudinally. Changes were evaluated for cadence; gait speed; step length; gait deviation index; kinematic data of the pelvis, hip, knee, and ankle joints; and foot progression angle.

RESULTS:

Levodopa treatment increased the cadence and gait speed in one of three patients and increased the gait deviation index in two of three patients. The kinematic data for each joint exhibited different characteristics, with some improvement observed in each of the three patients. There was consistent marked improvement in the abnormal foot progression angle; one patient had excessive external rotation of one foot, another had excessive bilateral internal rotation, and the other had excessive internal rotation of one foot and excessive external rotation of the opposite foot, all of which improved.

CONCLUSION:

The 3DGA findings demonstrate that the gait pathology and recovery process in GTPCH1-deficient DRD vary from case to case. Changes in the foot progression angle and gait deviation index can enable the effects of treatment to be more easily evaluated.
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Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Asunto principal: Levodopa / Trastornos Distónicos Límite: Humans Idioma: En Revista: Pediatr Neurol Asunto de la revista: NEUROLOGIA / PEDIATRIA Año: 2024 Tipo del documento: Article Pais de publicación: Estados Unidos
Texto completo
Añadir a Mi BVS
Imprimir
XML
PubMed Links
Buscar en Google

Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Asunto principal: Levodopa / Trastornos Distónicos Límite: Humans Idioma: En Revista: Pediatr Neurol Asunto de la revista: NEUROLOGIA / PEDIATRIA Año: 2024 Tipo del documento: Article Pais de publicación: Estados Unidos