Literature DB >> 21055769

Cor pulmonale in a patient with Brown-Vialetto-Van Laere syndrome: a case report.

Francisco Pereira da Silva-Júnior1, Rafael de Deus Moura, Sérgio Rosemberg, Paulo Eurípedes Marchiori, Luiz Henrique Martins Castro.   

Abstract

Brown-Vialetto-Van Laere syndrome (BVVLS) is a rare neurological disease characterized by sensorineural hearing loss and multiple cranial nerve palsies, usually involving the VIIth and IXth to XIIth cranial nerves. We describe the clinical and pathological features of a 33-year-old woman with BVVLS. The patient developed progressive exertional dyspnea, with clinical and laboratory findings of right-sided heart failure and pulmonary hypertension. She developed status epilepticus in the setting of cardiac deterioration and respiratory infection, and died of cardiogenic and septic shock. Autopsy disclosed bilateral neuronal loss and gliosis in the inferior colliculi, locus coeruleus and facial and vestibular nuclei. Cor pulmonale is a complication of hypoventilation-induced hypoxia and hypercapnia and had not yet been reported in BVVLS.
Copyright © 2010 Elsevier B.V. All rights reserved.

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Year:  2010        PMID: 21055769     DOI: 10.1016/j.jns.2010.10.010

Source DB:  PubMed          Journal:  J Neurol Sci        ISSN: 0022-510X            Impact factor:   3.181


  1 in total

Review 1.  The Brown-Vialetto-Van Laere and Fazio Londe syndrome revisited: natural history, genetics, treatment and future perspectives.

Authors:  Annet M Bosch; Kevin Stroek; Nico G Abeling; Hans R Waterham; Lodewijk Ijlst; Ronald J A Wanders
Journal:  Orphanet J Rare Dis       Date:  2012-10-29       Impact factor: 4.123

  1 in total

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