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Successful Treatment of Schwartz-Jampel Syndrome with Botulinum Toxin Type A

  • Panittra Suphatsathienkul
  • , Kullasate Sakpichaisakul
  • , Thanin Wechapinan
  • , Objoon Trachoo
  • , Sorawit Virawan
  • , Rungsima Wanitphakdeedecha
  • Siriraj Hospital
  • Queen Sirikit National Institute of Child Health
  • Rangsit University
  • Ministry of Public Health

Research output: Contribution to journalArticlepeer-review

1 Citation (Scopus)

Abstract

Schwartz-Jampel syndrome (SJS) is a rare autosomal recessive disorder characterized by typical facial dysmorphism, generalized muscle stiffness, joint contracture, and skeletal abnormalities. This condition is caused by mutations in the heparan sulfate proteoglycan 2 (HSPG2) gene, which encodes perlecan, a component of the basement membrane. The management of patients with SJS primarily aims to alleviate symptoms related to muscle stiffness. In this report, we describe a male patient with SJS type 1A. Trio whole-exome sequencing identified a pathogenic mutation (NM_001291860.1: c.10897C>T; p.Arg3633Ter) and variants of unknown significance (NM_001291860.2: c.413+10G>T). The patient experienced difficulty in opening his eyes and mouth, which significantly limited his daily activities. Botulinum toxin A injection was administered and demonstrated significant clinical improvement after the treatment.

Original languageEnglish
Pages (from-to)545-556
Number of pages12
JournalDermatology and Therapy
Volume14
Issue number2
DOIs
Publication statusPublished - Feb 2024

Keywords

  • Blepharophimosis
  • Botulinum toxin
  • HSPG2
  • Myotonia
  • Schwartz-Jampel syndrome

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