Korean J Crit Care Med.  2016 Feb;31(1):34-38. 10.4266/kjccm.2016.31.1.34.

Malignant Neuroleptic Syndrome following Deep Brain Stimulation Surgery of Globus Pallidus Pars Internus in Cerebral Palsy

Affiliations
  • 1Department of Neurosurgery, Seoul National University Hospital, Seoul, Korea. paeksh@snu.ac.kr
  • 2Department of Anesthesiology, Seoul National University Hospital, Seoul, Korea.
  • 3Department of Neurology, Seoul National University Hospital, Seoul, Korea.

Abstract

Neuroleptic malignant syndrome (NMS) is a rare but potentially lethal outcome caused by sudden discontinuation or dose reduction of dopaminergic agents. We report an extremely rare case of NMS after deep brain stimulation (DBS) surgery in a cerebral palsy (CP) patient without the withdrawal of dopaminergic agents. A 19-year-old girl with CP was admitted for DBS due to medically refractory dystonia and rigidity. Dopaminergic agents were not stopped preoperatively. DBS was performed uneventfully under monitored anesthesia. Dopaminergic medication was continued during the postoperative period. She manifested spasticity and muscle rigidity, and was high fever resistant to anti-pyretic drugs at 2 h postoperative. At postoperative 20 h, she suffered cardiac arrest and expired, despite vigorous cardiopulmonary resuscitation. NMS should be considered for hyperthermia and severe spasticity in CP patients after DBS surgery, irrespective of continued dopaminergic medication.

Keyword

cerebral palsy; deep brain stimulation; neuroleptic malignant syndrome

MeSH Terms

Anesthesia
Cardiopulmonary Resuscitation
Cerebral Palsy*
Deep Brain Stimulation*
Dopamine Agents
Dystonia
Female
Fever
Globus Pallidus*
Heart Arrest
Humans
Muscle Rigidity
Muscle Spasticity
Neuroleptic Malignant Syndrome
Postoperative Period
Young Adult
Dopamine Agents

Figure

  • Fig. 1. Axial (A) and coronal (B) postoperative computed tomography images. There was no intracranial abnormality and electrode leads were in the proper positions.

  • Fig. 2. An X-ray image shows no significant chest abnormality, however it does reveal abdominal air fluid levels suggesting ileus.

  • Fig. 3. A schematic illustration of changes in body temperature and the management of hyperthermia during the perioperative period. BT: body temperature; BP: blood pressure; HR: heart rate; RR: raspiration rate; R-ward: recovery ward; CPR: cardiopulmonary resuscitation; ICU: intensive care unit.


Reference

References

1. Vidailhet M, Yelnik J, Lagrange C, Fraix V, Grabli D, Thobois S, et al. Bilateral pallidal deep brain stimulation for the treatment of patients with dystonia-choreoathetosis cerebral palsy: a prospective pilot study. Lancet Neurol. 2009; 8:709–17.
Article
2. Jankovic J. Dystonia: medical therapy and botulinum toxin. Adv Neurol. 2004; 94:275–86.
3. Bhidayasiri R. Dystonia: genetics and treatment update. Neurologist. 2006; 12:74–85.
4. Kim AR, Chang JW, Chang WS, Park ES, Cho SR. Two-year outcomes of deep brain stimulation in adults with cerebral palsy. Ann Rehabil Med. 2014; 38:209–17.
Article
5. Adnet P, Lestavel P, Krivosic-Horber R. Neuroleptic malignant syndrome. Br J Anaesth. 2000; 85:129–35.
Article
6. Newman EJ, Grosset DG, Kennedy PG. The parkinsonism-hyperpyrexia syndrome. Neurocrit Care. 2009; 10:136–40.
Article
7. Kipps CM, Fung VS, Grattan-Smith P, de Moore GM, Morris JG. Movement disorder emergencies. Mov Disord. 2005; 20:322–34.
Article
8. Nicholson D, Chiu W. Neuroleptic malignant syndrome. Geriatrics. 2004; 59:36. 38-40.
9. American Psychiatric Association. Diagnostic and statistical manual of mental disorders: DSM-IV-TR. Washinton: American Psychiatric Association;2000. p. 795–8.
10. Levenson JL. Neuroleptic malignant syndrome. Am J Psychiatry. 1985; 142:1137–45.
11. Pranzatelli MR. Oral pharmacotherapy for the movement disorders of cerebral palsy. J Child Neurol. 1996; 11 Suppl 1:S13–22.
Article
12. DeLong M, Wichmann T. Deep brain stimulation for movement and other neurologic disorders. Ann N Y Acad Sci. 2012; 1265:1–8.
Article
13. Benabid AL, Benazzouz A, Hoffmann D, Limousin P, Krack P, Pollak P. Long-term electrical inhibition of deep brain targets in movement disorders. Mov Disord. 1998; 13 Suppl 3:119–25.
Article
14. Vercueil L, Krack P, Pollak P. Results of deep brain stimulation for dystonia: a critical reappraisal. Mov Disord. 2002; 17 Suppl 3:S89–93.
Article
15. Counelis GJ, Simuni T, Forman MS, Jaggi JL, Trojanowski JQ, Baltuch GH. Bilateral subthalamic nucleus deep brain stimulation for advanced PD: correlation of intraoperative MER and postoperative MRI with neuropathological findings. Mov Disord. 2003; 18:1062–5.
Article
16. Urasaki E, Fukudome T, Hirose M, Nakane S, Matsuo H, Yamakawa Y. Neuroleptic malignant syndrome (parkinsonism-hyperpyrexia syndrome) after deep brain stimulation of the subthalamic nucleus. J Clin Neurosci. 2013; 20:740–1.
Article
17. Perry D, Birthi P, Salles S, McDowell S. Neuroleptic malignant syndrome associated with the use of carbidopa/levodopa for dystonia in persons with cerebral palsy. PM R. 2012; 4:383–4.
Article
18. Sakkas P, Davis JM, Janicak PG, Wang ZY. Drug treatment of the neuroleptic malignant syndrome. Psychopharmacol Bull. 1991; 27:381–4.
19. Tse L, Barr AM, Scarapicchia V, Vila-Rodriguez F. Neuroleptic malignant syndrome: a review from a clinically oriented perspective. Curr Neuropharmacol. 2015; 13:395–406.
Article
Full Text Links
  • KJCCM
Actions
Cited
CITED
export Copy
Close
Share
  • Twitter
  • Facebook
Similar articles
Copyright © 2023 by Korean Association of Medical Journal Editors. All rights reserved.     E-mail: koreamed@kamje.or.kr