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Abrupt Intrathecal Baclofen Withdrawal: Management of Potentially Life-Threatening Sequelae

2001; Elsevier BV; Volume: 4; Issue: 4 Linguagem: Inglês

10.1046/j.1525-1403.2001.00142.x

1525-1403

Robert J. Coffey, Patrick M. Ridgely,

Neurological disorders and treatments

Intrathecal baclofen (ITB, Lioresal Intrathecal, Medtronic Neurological, Minneapolis, MN) delivered by an implanted programmable infusion pump is used increasingly to treat medically intractable spasticity and other centrally mediated hypertonic disorders ( 1. Albright AL Barry MJ Painter MJ Shultz B Infusion of intrathecal baclofen for generalized dystonia in cerebral palsy. J Neurosurg. 1998; 88: 73-76 Crossref PubMed Scopus (86) Google Scholar , 2. Albright AL Baclofen in the treatment of cerebral palsy. J Child Neurol. 1996; 11: 77-83 Crossref PubMed Scopus (130) Google Scholar , 3. Albright AL Cervi A Singletary J Intrathecal baclofen for spasticity in cerebral palsy. JAMA. 1991; 265: 1418-1422 Crossref PubMed Scopus (242) Google Scholar , 4. Butler C Campbell S Evidence of the effects of intrathecal baclofen for spastic and dystonic cerebral palsy. AACPDM Treatment Outcomes Committee Review Panel. Dev Med Child Neurol. 2000; 42 (DOI: 10.1017/s0012162200001183): 634-645 Crossref PubMed Scopus (176) Google Scholar , 5. Coffey RJ Cahill D Steers W et al. Intrathecal baclofen for intractable spasticity of spinal origin: results of a long-term multicenter study. J Neurosurg. 1993; 78: 226-232 Crossref PubMed Scopus (365) Google Scholar , 6. Gilmartin R Bruce D Storrs BB et al. Intrathecal baclofen for management of spastic cerebral palsy: multicenter trial. J Child Neurol. 2000; 15: 71-77 Crossref PubMed Scopus (206) Google Scholar , 7. Meythaler JM McCary A Hadley MN Prospective assessment of continuous intrathecal infusion of baclofen for spasticity caused by acquired brain injury: a preliminary report. J Neurosurg. 1997; 87: 415-419 Crossref PubMed Scopus (83) Google Scholar ). The dosage of ITB administered to each patient varies with his or her diagnosis, the length of treatment, and response to the drug. Although intrathecal drugs are effective, a human error or device malfunction can cause over or under dosage, or the cessation of drug infusion. ITB overdosage can cause rostral progression of hypotonia, respiratory depression, coma, and occasionally, seizures ( 8. Haubenstock A Hruby K Jager U Lenz K Baclofen intoxication: report of 4 cases and review of the literature. Clin Toxicol. 1983; 20: 59-68 Crossref Scopus (37) Google Scholar ). ITB withdrawal most often causes a return of the patient's baseline spasticity and rigidity ( 5. Coffey RJ Cahill D Steers W et al. Intrathecal baclofen for intractable spasticity of spinal origin: results of a long-term multicenter study. J Neurosurg. 1993; 78: 226-232 Crossref PubMed Scopus (365) Google Scholar ). However, abrupt withdrawal can cause generalized seizures, hallucinations, and symptoms that could be misdiagnosed as autonomic dysreflexia, malignant hyperthermia (MH), or neuroleptic-malignant syndrome (NMS) ( 9. 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Arch Neurol. 1981; 38: 588-589 Crossref PubMed Scopus (99) Google Scholar ). In a small fraction of patients ITB withdrawal has caused a life-threatening cascade of symptoms that consisted of exaggerated rebound spasticity, muscle rigidity, fever, labile blood pressure, and diminished level of consciousness. If not treated adequately, the syndrome progressed over 1–3 days to include rhabdomyolysis, elevated plasma creatine kinase (CK) levels, elevated transaminase levels, hepatic and renal failure, disseminated intravascular coagulation (DIC), and in some cases, death ( 21. Green LB Nelson VS Death after acute withdrawal of intrathecal baclofen: case report and literature review. Arch Phys Med Rehabil. 1999; 80: 1600-1604 Abstract Full Text PDF PubMed Scopus (98) Google Scholar , 22. Meinck H-M Tronnier V Reike K Wirtz CR Flügel D Schwab S Intrathecal baclofen treatment for stiff-man syndrome: pump failure may be fatal. Neurology. 1994; 44 (letter): 2209-2210 Crossref PubMed Google Scholar ). The authors, in cooperation with a panel of experienced clinicians, reviewed the pathophysiology of ITB withdrawal and the outcome of 16 severe cases reported in the medical literature or to the United States Food and Drug Administration (FDA) ( 23. Coffey RJ, Edgar TS, Francisco GE, et al. Abrupt withdrawal from intrathecal baclofen. Recognition and management of a potentially life–threatening syndrome. Arch Phys Med Rehabil 2001; in press Google Scholar ). Because adverse event reporting is voluntary, the total number of cases is unknown, and likely has been undercounted.