| Literature DB >> 34120822 |
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.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