Literature DB >> 34120822

Intracranial hemorrhage secondary to vitamin K deficiency in X-linked myotubular myopathy.

Jeremy M Neese1, Sabrina Yum2, Susan Matesanz3, Leslie J Raffini4, Hilary B Whitworth5, Kathleen M Loomes6, Oscar H Mayer7, Alicia M Alcamo8.   

Abstract

X-linked myotubular myopathy (XLMTM) is a rare congenital myopathy characterized by profound hypotonia and poor respiratory effort at birth. The condition is associated with multiple morbidities including chronic respiratory insufficiency, feeding tube dependence, and rarely, vitamin K deficiency leading to bleeding and coagulopathy. We report a case of a 6-month-old boy with X-linked myotubular myopathy who experienced a fatal intracranial hemorrhage due to vitamin K deficiency without prior clinical evidence of cholestasis or micronutrient deficiency. We propose clinically non-apparent cholestasis in combination with acute illness and poor weight gain led to his vitamin K deficiency and intracranial hemorrhage. However, the etiology and mechanism of his cholestasis remains unclear. We conclude that children with X-linked myotubular myopathy, especially with gene therapy on the horizon, may benefit from routine hepatic, coagulation, and nutritional screening to prevent potentially catastrophic bleeding.
Copyright © 2021 Elsevier B.V. All rights reserved.

Entities:  

Keywords:  Cholestasis; Coagulopathy; Intracranial hemorrhage; Vitamin K deficiency; X-linked myotubular myopathy (XLMTM)

Mesh:

Year:  2021        PMID: 34120822     DOI: 10.1016/j.nmd.2021.04.009

Source DB:  PubMed          Journal:  Neuromuscul Disord        ISSN: 0960-8966            Impact factor:   4.296


  2 in total

1.  INCEPTUS Natural History, Run-in Study for Gene Replacement Clinical Trial in X-Linked Myotubular Myopathy.

Authors:  James J Dowling; Wolfgang Müller-Felber; Barbara K Smith; Carsten G Bönnemann; Nancy L Kuntz; Francesco Muntoni; Laurent Servais; Lindsay N Alfano; Alan H Beggs; Deborah A Bilder; Astrid Blaschek; Tina Duong; Robert J Graham; Minal Jain; Michael W Lawlor; Jun Lee; Julie Coats; Charlotte Lilien; Linda P Lowes; Victoria MacBean; Sarah Neuhaus; Mojtaba Noursalehi; Teresa Pitts; Caroline Finlay; Sarah Christensen; Gerrard Rafferty; Andreea M Seferian; Etsuko Tsuchiya; Emma S James; Weston Miller; Bryan Sepulveda; Maria Candida Vila; Suyash Prasad; Salvador Rico; Perry B Shieh
Journal:  J Neuromuscul Dis       Date:  2022

2.  Intrahepatic Cholestasis Is a Clinically Significant Feature Associated with Natural History of X-Linked Myotubular Myopathy (XLMTM): A Case Series and Biopsy Report.

Authors:  Cristina Molera; Tinatin Sarishvili; Andrés Nascimento; Irakli Rtskhiladze; Gema Muñoz Bartolo; Santiago Fernández Cebrián; Justo Valverde Fernández; Beatriz Muñoz Cabello; Robert J Graham; Weston Miller; Bryan Sepulveda; Binita M Kamath; Hui Meng; Michael W Lawlor
Journal:  J Neuromuscul Dis       Date:  2022
  2 in total

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