Worsening spasticity due to catheter breakage during intrathecal baclofen therapy: a case report

Abstract

Background

Intrathecal baclofen therapy can substantially improve symptoms in patients with severe spasticity owing to traumatic spinal cord injury, multiple sclerosis, cerebral paresis, or tethered cord syndrome. Problems associated with intrathecal catheters include migration, laceration, occlusion, or disconnection. Several case reports have described the management of catheter fragments. To the best of our knowledge, this is the first detailed report of the insertion of a new catheter.

Case presentation

A 64-year-old Japanese man with spinal myoclonus was undergoing intrathecal baclofen therapy; his spasticity was well controlled with intrathecal baclofen therapy but worsened 13 years after pump implantation. Imaging revealed spinal catheter breakage, and the catheter was retained in the spinal canal. We inserted a new catheter through a different intervertebral space without removing the original catheter. Postoperatively, the spasticity remained well controlled with intrathecal baclofen therapy.

Conclusion

This is the first detailed report on the insertion of a new catheter for intrathecal baclofen therapy at a different intervertebral space from the catheter breakage, without removal of the old intrathecal catheter.

Background

Intrathecal baclofen (ITB) therapy can substantially improve symptoms in patients with severe spasticity owing to traumatic spinal cord injury, multiple sclerosis, cerebral paresis, or tethered cord syndrome [1,2,3,4,5,6,7,8]. The problems associated with ITB therapy include migration, laceration, occlusion, or disconnection [9]. Although there are several reports on retained catheter fragments and their management, they do not provide details on the specific surgical techniques in terms of the retained catheter [10,11,12,13,14,15,16,17]. To the best of our knowledge, detailed reports on the insertion of new catheters have also not yet been published. We report a case of insertion of a new catheter for intrathecal baclofen therapy at a different intervertebral space from catheter breakage, without the removal of the old intrathecal catheter.

Case presentation

A 64-year-old Japanese man had undergone cervical laminoplasty from C4 to C7 16 years prior. Although his myelopathy improved, he developed spasticity in both lower limbs and was diagnosed with myoclonus 14 years earlier. He had been referred to our department 13 years previously because oral medication had not alleviated his spasticity. He was employed and had no relevant family history, history of smoking tobacco, or alcohol consumption.

Laboratory assessments showed no signs of an inflammatory reaction: C-reactive protein, 0.08 mg/L; white blood cell count, 11.5 × 10⁹/L; and platelet count, 211 × 10⁹/L. In addition, laboratory assessments of liver and renal function showed no abnormal findings: aspartate aminotransferase (AST), 23 U/L; alanine aminotransferase (ALT), 25 U/L; alkaline phosphatase (ALP), 248 U/L (normal value 100–340); blood urea nitrogen (BUN), 18.1 mg/dl; and creatinine, 0.8 mg/dl. Urine, serological, and microbiological analyses revealed no abnormalities. His body temperature was 36.0 ˚C, pulse was 77 beats per minute, and blood pressure was 134/87 mmHg on admission. Physical findings showed that he had spasms in the upper and lower limbs, making it impossible for him to sleep supine, and he usually slept on his side with his hips bent. His lower legs would tap when he washed his hair, drove a car, or was startled. The degree of spasticity was evaluated using the Modified Ashworth scale (MAS) (Table 1) [18]. The MAS was evaluated for eight sites (left/right): hip abduction (2/1 +), hip flexion (2/1 +), knee flexion (2/1 +), and ankle extension (2/1 +) in both lower limbs and wrist flexion (2/1 +), wrist extension (2/1 +), elbow flexion (2/1 +), and elbow extension (2/1 +) in both upper limbs. To evaluate the effectiveness of ITB therapy, baclofen was administered intrathecally as a single 50 μg dose, resulting in complete resolution of his spasticity and myoclonus. The patient consented to the implantation of a programmable pump that could continuously deliver ITB. For implantation, a 16-gauge spinal needle was introduced into the L3/4 intervertebral space, under fluoroscopy, using the paramedian approach. After confirmation of the free flow of cerebrospinal fluid (CSF), the intrathecal catheter was passed through the needle, and the distal tip was placed at the level of T9 (Fig. 1). A pump for the continuous delivery of baclofen (SynchroMed II, Medtronic, Inc., Minneapolis, MN, USA) was implanted into the left abdomen. After ITB pump implantation, the spasticity improved immediately and significantly, and the myoclonus disappeared. The patient remained under excellent control on baclofen at 300–350 μg/day. Surgery was performed to replace the pump after 7 and 13 years. However, 1 week after the second pump replacement, the spasticity worsened. Despite initiating oral baclofen tablets and increasing the daily dose of baclofen using the pump, the patient’s spasticity did not improve. Computed tomography (CT) revealed that the spinal catheter was torn within the spinal canal between L3 and L4. The top of the intrathecal catheter was inserted at the T9 level, and fragments of the proximal and distal ends of the intrathecal catheter were observed at the L3/4 level (Fig. 2). Laboratory assessments showed no signs of an inflammatory reaction: C-reactive protein, 0.04 mg/L; white blood cell count , 7.6 × 10⁹/L; and platelet count, 197 × 10⁹/L. In addition, a laboratory assessment of liver and renal function showed no abnormal findings: AST, 30 U/L; ALT, 30 U/L; ALP [International Federation of Clinical Chemistry and Laboratory Medicine (IFCC)], 54 U/L (normal value 38–113); creatinine kinase (CK), 312 U/L; BUN, 17.7 mg/dl; creatinine, 0.85 mg/dl; sodium (Na), 140 mEq/L; potassium (K), 4.5, mEq/L; and chloride (Cl), 104 mEq/L. Blood test results for CK and potassium were within the normal range, and there were no clinical findings suggestive of withdrawal syndrome. The patients’ body temperature was 36.0 ˚C, pulse was 75 beats per minute, and blood pressure was 115/66 mmHg on admission. Physical examination revealed worsening spasms in the upper and lower extremities, making it difficult for the patient to walk independently. The MAS was evaluated for eight sites (left/right): hip abduction (2/1 +), hip flexion (2/1 +), knee flexion (2/1 +), and ankle extension (2/1 +) in both lower limbs and wrist flexion (2/1 +), wrist extension (2/1 +), elbow flexion (2/1 +), and elbow extension (2/1 +) in both upper limbs.

Table 1 Modified Ashworth Scale for the grading of spasticity

Fig. 1

Intrathecal baclofen pump implantation. Intrathecal baclofen pump implantation was performed by inserting an intrathecal catheter through the L3/4 intervertebral space (white solid triangle) to the T9 level (white outline arrow) (a anterior–posterior (AP) view; b lateral view)

Fig. 2

Computed tomography of the thoracolumbar spine. The distal tip of the intrathecal catheter was introduced at the T9 level (white outlined arrow), and breakage of the intrathecal catheter was observed at the L3/4 level (white solid arrow) (a sagittal view; b L3/4 axial view; c L3/4 distal axial view; d L4 proximal axial view). Fragments of the proximal and distal ends of the intrathecal catheter were observed at the L3/4 level; e three-dimensional computed tomography scan

We discussed, with the patient and his wife, whether to remove the remaining catheter and insert a new catheter or to leave the remaining catheter as is and insert a new catheter. They decided to leave the original catheter in place and insert a new one.

During surgery, the patient was placed in the right lateral position under general anesthesia by an anesthetist. A skin incision was made proximal to the site of the previous incision. For implantation, a 16-gauge spinal needle was introduced into the L2/3 intervertebral space under fluoroscopy, using the paramedian approach, taking care not to damage the original catheter. After confirming free flow of CSF, the Ascenda catheter was passed through the needle. Fluoroscopy confirmed that it had ascended smoothly. The distal tip of the Ascenda catheter was placed at the same T9 position as the previous catheter (Fig. 3). The torn spinal catheter was removed from the soft tissue distal to the insertion site of the new catheter. The pump end of the catheter was cut, and the old catheter was removed. The original spinal catheter was left in place in the spinal canal. The new catheter was connected to the pump to allow the baclofen to reach the subarachnoid space (Fig. 4). Postoperatively, a new intrathecal catheter was introduced through the L2/3 intervertebral space to the original position at the T9 level (Fig. 5). After ITB pump implantation, the patients’ spasticity greatly improved and he was able to walk without assistance. The MAS at the eight sites were hip extensors (1/1), hip adductors (1/1), knee extensors (1/1), and ankle plantar flexors (1/1). During the 5-month follow-up, spasticity remained under excellent control on baclofen at 160–170 μg/day without adverse effects. Radiographic examination performed 5 months after surgery showed no migration of the original catheter or the newly inserted catheter.

Fig. 3

Intraoperative image. A new intrathecal catheter was introduced through the L2/3 intervertebral space to the original T9 level (new catheter, black solid arrow; old catheter, black outline arrow) (a anterior−posterior view; b lateral view; c lateral view; d lateral view)

Fig. 4

Intraoperative findings and the extracted intrathecal catheter. The spinal segment of the intrathecal catheter was removed (torn spinal catheter, white arrow; new spinal segment of the intrathecal catheter, white solid triangle; new pump segment of the intrathecal catheter, white outline triangle). We extracted the torn intrathecal catheter (white arrow) (a intraoperative findings; b extracted intrathecal catheter)

Fig. 5

Postoperative X-ray. A new intrathecal catheter was introduced through the L2/3 intervertebral space to the original T9 level (new catheter, white solid arrow; old catheter, white outline arrow) (a anterior−posterior view; b lateral view)

Discussion and conclusion

ITB therapy is an effective treatment for the management of spasticity of different origins [1,2,3,4,5,6,7,8]; however, complications related to the device or administered drug may occur and must be managed [9,10,11,12,13,14,15,16,17]. Problems associated with intrathecal catheters include migration, laceration, occlusion, and disconnection, which cause sudden cessation of intrathecal drug administration [9]. The Ascenda catheter features a unique four-layer design to reduce catheter-related problems such as collapse, kinking, and occlusion [16]. Data published in the Medtronic Product Performance Report summary from the Medtronic post-market registry 2022 (version 1.0, 20 March) show that the InDura catheter was approved by the Food and Drug Administration (FDA) in October 1999. Among the 661 patients fitted with the InDura catheter, catheter break/cut has been observed in 19 patients (2.8%). The Ascenda catheter was approved by the FDA in May 2012. Among the 1256 patients using the Ascenda catheter, catheter break/cut has been observed in 6 patients (0.48%).

Frizon et al. [10] reported the results of a retrospective review of 59 patients, previously implanted with intrathecal drug delivery systems, who underwent intrathecal catheter removal. In total, 8 of the 59 patients (13.5%) experienced complications. A retained catheter was the most common complication, occurring in four out of eight patients (50%). Of these four patients, two required further surgery to remove the retained portion of the intrathecal catheter. One patient developed a compressive subdural fluid collection around the broken tip at the T10–T11 level. The other patient had a retained intradural and extradural catheter piece that was subsequently removed after recurrent infection. CSF leak was the second complication, occurring in three out of eight patients (37.5%). Of these three patients, two required an external drain to divert the CSF, and one improved with conservative management. Persistent headache was the third complication, occurring in one out of eight patients (12.5%). This patient required three blood patches to improve their symptoms.

Vodapally et al. [11] reported a case of a tension pseudomeningocele caused by a retained lumbar intrathecal catheter. The authors stated that if the spinal end of the catheter is retracted deep into the interspinous ligaments and is not recoverable, entering the intrathecal space at the same level should be avoided.

Nakaji et al. [12] reported a case of cranial subarachnoid hemorrhage (SAH) related to the cephalad migration of an indwelling intrathecal catheter within the spinal canal, which was extracted from below through C6–C7 laminoplasty without complications.

Hnenny et al. [13] reported an unusual case of a patient presenting with a posterior fossa subarachnoid hemorrhage (SAH) associated with the fracture and migration of a narcotic pump lumbar intrathecal catheter into the upper cervical subarachnoid space and infratentorial cisterns.

Maugans [14] reported a case of intracranial extension of a catheter segment. The segment was removed from the subarachnoid space, there were no perioperative complications, and the symptoms of neck pain, dizziness, and nausea resolved.

Oshino et al. [15] published a technical note describing a practical approach for removing a sheared lumbar catheter fragment and avoiding its migration into the spinal canal.

Although several reports on retained catheter fragments and their management have been published, they do not provide details on the specific surgical techniques used to deal with the retained catheter. The authors also do not provide details on the implantation of a new intrathecal catheter.

To remove an intrathecal foreign body, invasive surgeries (such as laminectomy or laminotomy) are necessary. Therefore, risks and benefits should be assessed prior to decision-making in these cases [17]. During surgery, we were careful to insert the new catheter into the intervertebral space one level above, rather than from the same intervertebral space as the previous one, to avoid a CSF fluid fistula and damage to the original catheter. A 16-gauge spinal needle was inserted under fluoroscopic guidance. To our knowledge, this is the first detailed report on the insertion of a new catheter for ITB therapy at a different intervertebral space from the catheter breakage, without removal of the old intrathecal catheter. With the new catheter inserted, the spasticity is well controlled 5 months after surgery. Since the old intrathecal catheter remains in place, attention must be paid to catheter migration, and long-term follow-up is required.

Conclusion

We report a case of insertion of a new catheter for ITB therapy at a different intervertebral space from the catheter breakage, without the removal of the old intrathecal catheter. We considered that insertion of a new catheter into a different intervertebral space would be a useful alternative for the management of catheter breakage.