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Recovery in a Patient with Locked-In Syndrome

Published online by Cambridge University Press:  02 December 2014

Brian Silver ,
Kavita M. Grover ,
Ximena Arcila ,
Panayiotis D. Mitsias ,
Susan M. Bowyer  and
Michael Chopp
Brian Silver*
Affiliation:
Department of Neurology, Henry Ford Health System, Detroit, MI Wayne State University, Detroit, MI
Kavita M. Grover
Affiliation:
Department of Neurology, Henry Ford Health System, Detroit, MI
Ximena Arcila
Affiliation:
Department of Neurology, Henry Ford Health System, Detroit, MI Wayne State University, Detroit, MI
Panayiotis D. Mitsias
Affiliation:
Department of Neurology, Henry Ford Health System, Detroit, MI Wayne State University, Detroit, MI
Susan M. Bowyer
Affiliation:
Department of Neurology, Henry Ford Health System, Detroit, MI Oakland University, Rochester, MI, USA
Michael Chopp
Affiliation:
Department of Neurology, Henry Ford Health System, Detroit, MI Oakland University, Rochester, MI, USA
*
Department of Neurology, Henry Ford Hospital, 2799 West Grand Boulevard, K-11, Detroit, Michigan, 48202, USA
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Abstract:

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Background and Purpose:

Sildenafil citrate has been shown to enhance neurogenesis, angiogenesis, synaptogenesis, and neurological outcome by augmentation of cyclic guanosine monophosphate (cGMP) levels in animal models of ischemic stroke. Whether sildenafil citrate may be helpful for recovery in human stroke is unknown at this time.

Methods:

A 41-year-old woman with locked-in syndrome due to pontine infarction began receiving 150 mg of oral sildenafil citrate daily on a compassionate use basis in August 2003 and continues treatment at this time. Magnetoencephalography (MEG) was performed at 12 and 17 months after stroke.

Results:

No serious adverse events have occurred. Significant milestone recoveries including standing, use of both arms, talking, and full return of swallowing have occurred, particularly after nine months of treatment. The MEG showed a significantly increased amplitude in the somatosensory cortex.

Conclusion:

Daily use of high dose sildenafil citrate appears to be safe in this patient with stroke resulting in locked-in syndrome. Further studies will be required to establish safety and efficacy.

Résumé:

RÉSUMÉ:Contexte et objectif:

La neurogenèse, l’angiogenèse et la synaptogenèse sont stimulées par le citrate de sildenafil et l’issue neurologique est améliorée à cause de l’augmentation des niveaux de guanosine monophosphate cyclique (cGMP) chez des modèles animaux d’accident vasculaire cérébral ischémique. On ne sait pas si le citrate de sildenafil peut être utile dans la récupération d’un accident vasculaire cérébral chez l’humain.

Méthodes:

Une femme âgée de 41 ans, atteinte du locked-in syndrome dû à un infarctus du pont, a commencé à recevoir 150 mg de citrate de sildenafil per os quotidiennement en août 2003 (accès spécial). Elle prend toujours cette médication. Une magnéto-encéphalographie (MEG) a été effectuée 12 et 17 mois après l’infarctus.

Résultats:

Elle n’a présenté aucun incident thérapeutique. Des étapes importantes au point de vue récupération ont été franchies : la station debout, l’utilisation des deux bras, la parole et la déglutition. Ceci était plus évident après neuf mois de traitement. On a observé une augmentation significative de l’amplitude dans le cortex somesthésique à la MEG.

Conclusion:

La prise quotidienne d’une dose élevée de citrate de sildenafil semble sûre chez cette patiente atteinte du locked-in syndrome suite à un accident vasculaire cérébral. Sa sécurité et son efficacité devront toutefois faire l’objet d’études plus poussées

Type
Case Report
Information
Canadian Journal of Neurological Sciences , Volume 33 , Issue 2 , May 2006 , pp. 246 - 249
Copyright
Copyright © The Canadian Journal of Neurological 2006

References

1. Smith, E, Delargy, M. Locked-in syndrome. Br Med J. 2005;330:406–9.Google Scholar
2. Leon-Carrion, J, van Eeckhout, P, Dominguez-Morales Mdel, R, Perez-Santamaria, FJ. The locked-in syndrome: a syndrome looking for a therapy. Brain Inj. 2002;16:571–82.Google Scholar
3. Patterson, JR, Grabois, M. Locked-in syndrome: a review of 139 cases. Stroke. 1986;17:758–64.Google Scholar
4. Casanova, E, Lazzari, RE, Lotta, S, Mazzucchi, A. Locked-in syndrome: improvement in the prognosis after an early intensive multidisciplinary rehabilitation. Arch Phys Med Rehabil. 2003;84:862–7.CrossRefGoogle ScholarPubMed
5. Gould, E, Reeves, AJ, Graziano, MS, Gross, CG. Neurogenesis in the neocortex of adult primates. Science. 1999;286:548–52.Google Scholar
6. Zhang, R, Wang, Y, Zhang, L, Zhang, Z, Tsang, W, Lu, M, et al. Sildenafil (Viagra) induces neurogenesis and promotes functional recovery after stroke in rats. Stroke. 2002;33:2675–80.CrossRefGoogle ScholarPubMed
7. Zhang, R, Wang, L, Zhang, L, Chen, J, Zhu, Z, Zhang, Z, et al. Nitric oxide enhances angiogenesis via the synthesis of vascular endothelial growth factor and cGMP after stroke in the rat. Circ Res. 2003;92:308–13.Google Scholar
8. Zhang, L, Zhang, RL, Wang, Y, Zhang, C, Zhang, ZG, Meng, H, et al. Functional recovery in aged and young rats after embolic stroke: treatment with a phosphodiesterase type 5 inhibitor. Stroke. 2005;36:847–52.Google Scholar
9. Rossini, PM, Tecchio, F, Pizzella, V, Lupoi, D, Cassetta, E, Pasqualetti, P. Interhemispheric differences of sensory hand areas after monohemispheric stroke: MEG/MRI integrative study. Neuroimage. 2001;14:474–85.Google Scholar
10. Gallien, P, Aghulon, C, Durufle, A, Petrilli, S, de Crouy, AC, Carsin, M, et al. Magnetoencephalography in stroke: a 1-year follow-up study. Eur J Neurol. 2003;10:373–82.Google Scholar
11. Moran, JE, Bowyer, SM, Tepley, N. Multi-Resolution FOCUSS: a source imaging technique applied to MEG data. Brain Topogr. 2005;18:1–17.CrossRefGoogle ScholarPubMed
12. Haig, AJ, Katz, RT, Sahgal, V. Mortality and complications of the locked-in syndrome. Arch Phys Med Rehabil. 1987;68:24–7.Google ScholarPubMed
13. McCusker, EA, Rudick, RA, Honch, GW, Griggs, RC. Recovery from the ‘locked-in’ syndrome. Arch Neurol. 1982;39:145–7.Google Scholar
14. Khurana, RK, Genut, AA, Yannakakis, GD. Locked-in syndrome with recovery. Ann Neurol. 1980;8:439–41.CrossRefGoogle ScholarPubMed