Ann Rehabil Med.  2015 Apr;39(2):308-312. 10.5535/arm.2015.39.2.308.

Paroxysmal Autonomic Instability With Dystonia Managed Using Chemodenervation Including Alcohol Neurolysis and Botulinum Toxin Type A Injection: A Case Report

• Lee HS
• Oh HS
• Shin JH

• KMID: 2273054
• DOI: http://doi.org/10.5535/arm.2015.39.2.308

Abstract

Paroxysmal autonomic instability with dystonia (PAID) is a rare complication of brain injury. Symptoms of PAID include diaphoresis, hyperthermia, hypertension, tachycardia, and tachypnea accompanied by hypertonic movement. Herein, we present the case of a 44-year-old female patient, who was diagnosed with paraneoplastic limbic encephalopathy caused by thyroid papillary cancer. The patient exhibited all the symptoms of PAID. On the basis that the symptoms were unresponsive to antispastic medication and her liver function test was elevated, we performed alcohol neurolysis of the musculocutaneous nerve followed by botulinum toxin type A (BNT-A) injection into the biceps brachii and brachialis. Unstable vital signs and hypertonia were relieved after chemodenervation. Accordingly, alcohol neurolysis and BNT-A injection are proposed as a treatment option for intractable PAID.

Keyword

Paroxysmal autonomic instability with dystonia; Botulinum toxins; Nerve block; Dystonia; Autonomic nervous system

MeSH Terms

Adult
Autonomic Nervous System
Botulinum Toxins
Botulinum Toxins, Type A*
Brain Injuries
Dystonia*
Female
Fever
Humans
Hypertension
Liver Function Tests
Musculocutaneous Nerve
Nerve Block*
Tachycardia
Tachypnea
Thyroid Gland
Vital Signs
Botulinum Toxins
Botulinum Toxins, Type A

Figure

• 

  Fig. 1 (A) Initial elbow flexed posture. (B) Ultrasound-guided musculocutaneous neurolysis with alcohol (solid arrow, needle; dashed arrow, musculocutaneous nerve). (C) Extended elbow posture after alcohol neurolysis.

• 

  Fig. 2 Change in vital signs with alcohol neurolysis and botulinum toxin type A (BTN-A) injection. (A) Body temperature, (B) systolic blood pressure, (C) pulse rate, and (D) respiration rate.

Reference

1. Blackman JA, Patrick PD, Buck ML, Rust RS Jr. Paroxysmal autonomic instability with dystonia after brain injury. Arch Neurol. 2004; 61:321–328. PMID: 15023807.

2. Ramdhani NA, Sikma MA, Witkamp TD, Slooter AJ, de Lange DW. Paroxysmal autonomic instability with dystonia in a patient with tuberculous meningitis: a case report. J Med Case Rep. 2010; 4:304. PMID: 20831807.

3. Comella CL, Leurgans S, Wuu J, Stebbins GT, Chmura T. Dystonia Study Group. Rating scales for dystonia: a multicenter assessment. Mov Disord. 2003; 18:303–312. PMID: 12621634.

4. Boeve BF, Wijdicks EF, Benarroch EE, Schmidt KD. Paroxysmal sympathetic storms ("diencephalic seizures") after severe diffuse axonal head injury. Mayo Clin Proc. 1998; 73:148–152. PMID: 9472998.

5. Sneed RC. Hyperpyrexia associated with sustained muscle contractions: an alternative diagnosis to central fever. Arch Phys Med Rehabil. 1995; 76:101–103. PMID: 7811167.

6. Goh KY, Conway EJ, DaRosso RC, Muszynski CA, Epstein FJ. Sympathetic storms in a child with a midbrain glioma: a variant of diencephalic seizures. Pediatr Neurol. 1999; 21:742–744. PMID: 10580889.

7. Thorley RR, Wertsch JJ, Klingbeil GE. Acute hypothalamic instability in traumatic brain injury: a case report. Arch Phys Med Rehabil. 2001; 82:246–249. PMID: 11239318.

8. Cuny E, Richer E, Castel JP. Dysautonomia syndrome in the acute recovery phase after traumatic brain injury: relief with intrathecal Baclofen therapy. Brain Inj. 2001; 15:917–925. PMID: 11595088.

9. Goddeau RP Jr, Silverman SB, Sims JR. Dexmedetomidine for the treatment of paroxysmal autonomic instability with dystonia. Neurocrit Care. 2007; 7:217–220. PMID: 17603760.

10. Taylor JJ, Woolsey RM. Dilute ethyl alcohol: effect on the sciatic nerve of the mouse. Arch Phys Med Rehabil. 1976; 57:233–237. PMID: 179494.