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Table 1 Summary of reported cases of ITB withdrawal

From: Status dystonicus resembling the intrathecal baclofen withdrawal syndrome: a case report and review of the literature

Author/Reference Patient and Primary Diagnosis Reason for ITB failure Features Reported (those in italics were not a feature of the case we report) Management and Outcome
Hansen et al. [12] 11 F Cerebral Palsy Removal of infected ITB Pump several hours previously Dystonia, Pyrexia, Rhabdomyolysis, Tachycardia, Hypertension, Reduced O2 Saturation, Diaphoresis Admitted to ICU and continuously sedated. Oral baclofen, dantrium, cyproheptadine, propofol and clonidine. Pump replaced with good result.
Samson-Fang et al. [7] 9 M Cerebral Palsy Disconnection of distal tubing from proximal segment. Time to onset unknown. Dystonia, Pyrexia (39.5°C), Rhabdomyolysis (15,200 IU/L), Agitation, Hypertension, Seizure, Scrotal and Ankle Oedema, Clotting Derangement, Metabolic Acidosis, Transaminase Elevation, Leukocytosis, Renal Failure Oral baclofen and clorazepate. Pump system revised. Patient subsequently deferevesced and tone returned to baseline.
Kao et al. [4] 4 M Cerebral Palsy Pump noted to be empty and beeping. Time to onset unknown. Dystonia, Pyrexia (42.1°C), Tachycardia, Tachypnoea, Hypertension, Reduced O2 Saturation, Wheeze, Crepitations, Leukocytosis Admitted to PICU. Paracetamol and bronchodilators. Following pump refill, fever and tachycardia resolved over four to eight hours. Recovery.
Salazar et al. [13] 14 M Cerebral Palsy Less than 1 mL left in pump reservoir Dystonia, Rhabdomyolysis (2500 IU/L), Tachycardia, Agitation, Hypertension, Diaphoresis, Haematemesis, Melaena, Clotting Derangement, Transaminase Elevation Pump refilled. Diazepam and cyproheptadine used to control tone. Discharged on hospital day four.
Saveika et al. [14] 14 F Cerebral Palsy Pump Stall 8 hrs previously Dystonia, Pyrexia (37.8°C), Tachycardia, Agitation, Pruritis Oral baclofen and cyproheptadine. Recovery within 24 hours.
Alden et al. [5] 14 M Adrenoleukodystrophy Catheter failure 1 week following spinal surgery Dystonia, Pyrexia (40°C), Tachypnoea IV lorazepam and oral baclofen. Pump and catheter revision. Discharged on post-operative day two.
Zuckerbraun et al. [15] 7 F Traumatic Brain Injury Due for refill the following day Dystonia, Pyrexia (40.8°C), Tachycardia, Impaired Consciousness, Tachypnoea, Diaphoresis, Seizure, Leukocytosis Baclofen bolus and pump refill. Defervesced over four hours.
Zuckerbraun et al. [15] 14 M Traumatic Brain Injury ITB catheter disconnection. Time to onset unknown. Dystonia, Pyrexia (41°C), Tachycardia, Agitation, Hypotension Four day admission. Misdiagnosed with viral illness and discharged. Withdrawal episode diagnosed on the basis of a subsequently identified catheter disconnection. Catheter subsequently revised with an improvement in tone.