title: Longitudinal Patterns of Thalidomide Neuropathy in Children and Adolescents.
abstract: OBJECTIVE
  To characterize the longitudinal clinical and electrophysiological patterns of thalidomide neuropathy in children and adolescents.
  
  
  METHODS
  Retrospective analysis of clinical records at a tertiary care children's hospital, including serial electrophysiological studies.
  
  
  RESULTS
  Sixteen patients aged 6-24 years received thalidomide to treat Crohn's disease from 2002 to 2012. Nine subjects had electrophysiological evidence of sensorimotor axonal polyneuropathy, 8 of whom had sensory and/or motor symptoms. The patients with polyneuropathy received thalidomide for 5 weeks to 52 months, with cumulative doses ranging from 1.4 to 207.7 g. All subjects with cumulative doses greater than 60 g developed polyneuropathy, and 4 of the 5 subjects who received thalidomide for more than 20 months developed polyneuropathy. The 7 subjects who had normal neurophysiological studies received therapy for 1 week to 25 months, with cumulative doses ranging from 0.7 to 47 g. In contrast to some previous reports, several patients had sensorimotor polyneuropathies, rather than pure sensory neuropathies. In patients with neuropathy who received therapy for more than 24 months and had 3 or more electromyography studies, the severity of the neuropathy plateaued.
  
  
  CONCLUSIONS
  Factors in addition to the total dose may contribute to the risk profile for thalidomide neuropathy, including pharmacogenetic susceptibilities. The severity of the neuropathy does not worsen relentlessly. Children, adolescents, and young adults receiving thalidomide should undergo regular neurophysiological studies to monitor for neuropathy.
authors: Department of Neurology, Boston Children's Hospital and Harvard Medical School, Boston, MA; Neurology Service, Department of Pediatrics, KK Women's and Children's Hospital, Singapore.
authors: Wendy K M
authors: WK
authors: Liew
authors: Child Health Research Institute, Department of Pediatrics, University of Florida College of Medicine, Gainesville, FL.
authors: Christina A
authors: CA
authors: Pacak
authors: Department of Neurology, Boston Children's Hospital and Harvard Medical School, Boston, MA.
authors: Nicole
authors: N
authors: Visyak
authors: Department of Neurology, Boston Children's Hospital and Harvard Medical School, Boston, MA.
authors: Basil T
authors: BT
authors: Darras
authors: Division of Gastroenterology, Hepatology, and Nutrition, Boston Children's Hospital and Harvard Medical School, Boston, MA.
authors: Athos
authors: A
authors: Bousvaros
authors: Department of Neurology, Boston Children's Hospital and Harvard Medical School, Boston, MA; Division of Pediatric Neurology, Department of Pediatrics, University of Florida College of Medicine, Gainesville, FL. Electronic address: pbkang@ufl.edu.
authors: Peter B
authors: PB
authors: Kang
mesh_terms: D000293:Adolescent; D002648:Child; D003424:Crohn Disease / Q000188:drug therapy*; D005260:Female; D006801:Humans; D007166:Immunosuppressive Agents / Q000009:adverse effects*; D008137:Longitudinal Studies; D008297:Male; D009431:Neural Conduction; D010523:Peripheral Nervous System Diseases / Q000139:chemically induced* / Q000453:epidemiology; D012189:Retrospective Studies; D013792:Thalidomide / Q000009:adverse effects*; D055815:Young Adult
chemical_list: D007166:Immunosuppressive Agents; D013792:Thalidomide
keywords: Crohn disease; sensorimotor polyneuropathy; thalidomide