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Stormorken Syndrome: A Rare Cause of Myopathy With Tubular Aggregates and Dystrophic Features.
Li, Ang; Kang, Xuan; Edelman, Frederick; Waclawik, Andrew J.
Afiliación
  • Li A; 1 Department of Neurology, University of Wisconsin School of Medicine and Public Health, Madison, WI, USA.
  • Kang X; 1 Department of Neurology, University of Wisconsin School of Medicine and Public Health, Madison, WI, USA.
  • Edelman F; 1 Department of Neurology, University of Wisconsin School of Medicine and Public Health, Madison, WI, USA.
  • Waclawik AJ; 1 Department of Neurology, University of Wisconsin School of Medicine and Public Health, Madison, WI, USA.
J Child Neurol ; 34(6): 321-324, 2019 05.
Article en En | MEDLINE | ID: mdl-30761937
Stormorken syndrome is a rare genetic disorder (MIM 185070) first reported in 1983 with thrombocytopenia, muscle weakness, asplenia, and miosis caused by a mutation of the stromal interaction molecule 1 ( STIM1) gene.1 The muscle weakness is caused by a myopathy with tubular aggregate formation. We report a family in which both child and mother presented with proximal muscle weakness and thrombocytopenia. Histologic, histochemical, and electron microscopy studies were performed on the muscle specimen. It documented accumulation of tubular aggregates and chronic myopathic changes with dystrophic features. Genetic testing revealed that both mother and son carried a missense mutation of c.326A>G in exon 3 of the STIM1 gene, which is novel for Stormorken syndrome. We suggest that patients with unexplained chronic idiopathic thrombocytopenia and proximal weakness have genetic testing for Stormorken syndrome.
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Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Asunto principal: Bazo / Trastornos de las Plaquetas Sanguíneas / Miosis / Dislexia / Ictiosis / Trastornos Migrañosos Tipo de estudio: Diagnostic_studies / Etiology_studies Límite: Child, preschool / Humans / Male Idioma: En Revista: J Child Neurol Asunto de la revista: NEUROLOGIA / PEDIATRIA Año: 2019 Tipo del documento: Article País de afiliación: Estados Unidos Pais de publicación: Estados Unidos

Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Asunto principal: Bazo / Trastornos de las Plaquetas Sanguíneas / Miosis / Dislexia / Ictiosis / Trastornos Migrañosos Tipo de estudio: Diagnostic_studies / Etiology_studies Límite: Child, preschool / Humans / Male Idioma: En Revista: J Child Neurol Asunto de la revista: NEUROLOGIA / PEDIATRIA Año: 2019 Tipo del documento: Article País de afiliación: Estados Unidos Pais de publicación: Estados Unidos