Literature DB >> 12569706

[Stormorken's syndrome].

Helge Stormorken1.   

Abstract

In 1985, a new syndrome with the following characteristics was described: thrombopathia, thrombopenia, asplenia, miosis, headache, ichthyosis, dyslexia, muscle defect, and subsequently also hypocalcaemia. Skin and deep bleedings, leg spasms, disturbed dark vision and dyslexia are main worries. This paper describes these patients with a review of the investigations performed. Causes of the bleeding tendency are complex disturbances of the platelet membrane causing insufficient stability of the haemostatic plug, the nature of which is unresolved, but involves membrane scrambling. The muscle defect consists in tubular aggregates and high blood values of creatine kinase. A connection with the hypocalcaemia is possible, because increasing the ionic Ca with calcitriol significantly improves muscle function. Miosis is resistant to mydriatics and causes decreased dark vision, possibly also influencing dyslexia. The asplenia has little influence on immunocompetence, and the patients have survived 300 patient years without critical infections. The gene defect has not yet been unravelled.

Entities:  

Mesh:

Year:  2002        PMID: 12569706

Source DB:  PubMed          Journal:  Tidsskr Nor Laegeforen        ISSN: 0029-2001


  4 in total

Review 1.  Role of STIM1/ORAI1-mediated store-operated Ca2+ entry in skeletal muscle physiology and disease.

Authors:  Antonio Michelucci; Maricela García-Castañeda; Simona Boncompagni; Robert T Dirksen
Journal:  Cell Calcium       Date:  2018-10-30       Impact factor: 6.817

2.  Activating mutations in STIM1 and ORAI1 cause overlapping syndromes of tubular myopathy and congenital miosis.

Authors:  Vasyl Nesin; Graham Wiley; Maria Kousi; E-Ching Ong; Thomas Lehmann; David J Nicholl; Mohnish Suri; Nortina Shahrizaila; Nicholas Katsanis; Patrick M Gaffney; Klaas J Wierenga; Leonidas Tsiokas
Journal:  Proc Natl Acad Sci U S A       Date:  2014-03-03       Impact factor: 11.205

3.  Stormorken Syndrome Caused by a p.R304W STIM1 Mutation: The First Italian Patient and a Review of the Literature.

Authors:  Oscar Borsani; Daniela Piga; Stefania Costa; Alessandra Govoni; Francesca Magri; Andrea Artoni; Claudia M Cinnante; Gigliola Fagiolari; Patrizia Ciscato; Maurizio Moggio; Nereo Bresolin; Giacomo P Comi; Stefania Corti
Journal:  Front Neurol       Date:  2018-10-15       Impact factor: 4.003

4.  Control of PTH secretion by the TRPC1 ion channel.

Authors:  Marta Onopiuk; Bonnie Eby; Vasyl Nesin; Peter Ngo; Megan Lerner; Caroline M Gorvin; Victoria J Stokes; Rajesh V Thakker; Maria Luisa Brandi; Wenhan Chang; Mary Beth Humphrey; Leonidas Tsiokas; Kai Lau
Journal:  JCI Insight       Date:  2020-04-23
  4 in total

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