INTRODUCTION: Intrathecal baclofen, administered via a Baclofen pump, is used for patients with spasticity. We report here a case of intrathecal catheter retraction following surgery. CASE REPORT: A male patient with adrenoleukodystrophy and a baclofen pump implant was admitted to the urology department with bladder stones. A transurethral cystolithotripsy and a suprapubic cystotomy were performed. Following surgery there was no adequate spasm control. Plain abdominal X-ray showed complete retraction of the catheter out of the intrathecal space. DISCUSSION: Intrathecal catheter retraction after surgery has been reported in a few cases. The retraction in this case did not seem to be due to the suprapubic cystotomy itself, but was more likely due to the positioning of the patient for cystoscopy and surgery, combined with possible loosening of the anchoring of the catheter. CONCLUSION: Specific attention should be paid to the positioning of patients before surgery in order to avoid intrathecal baclofen catheter withdrawal.