Recurrent abdominal dislodgement of intrathecal pump and utility of infraclavicular site for patients with elevated body mass index: illustrative case

Kristin Buxton Baclofen Pump Program, Boston Children’s Hospital, Boston, Massachusetts; and

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Ann Morgan Baclofen Pump Program, Boston Children’s Hospital, Boston, Massachusetts; and

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Weston T. Northam Department of Neurosurgery Boston Children’s Hospital, Boston, Massachusetts

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Scellig S. D. Stone Department of Neurosurgery Boston Children’s Hospital, Boston, Massachusetts

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BACKGROUND

Intrathecal baclofen, delivered via implanted pump, has been used to manage spasticity for approximately 40 years. The device is typically subcutaneously or subfascially implanted in the abdominal wall. There are very few cases reported of the pump being implanted in other locations.

OBSERVATIONS

This case describes the complicated course of a patient presenting with multiple episodes of catheter malfunction related to pump flipping in the abdominal pocket. The patient was successfully treated with repositioning of the pump into the infraclavicular fossa.

LESSONS

Infraclavicular placement of the implanted pump allowed for a more secure pocket base for this patient and less strain applied to the pump, minimizing the risk of disruption of pump positioning and interruption of drug delivery.

ABBREVIATIONS

CP = cerebral palsy; ITB = intrathecal baclofen; GABA = γ-amino-butyric-acid

BACKGROUND

Intrathecal baclofen, delivered via implanted pump, has been used to manage spasticity for approximately 40 years. The device is typically subcutaneously or subfascially implanted in the abdominal wall. There are very few cases reported of the pump being implanted in other locations.

OBSERVATIONS

This case describes the complicated course of a patient presenting with multiple episodes of catheter malfunction related to pump flipping in the abdominal pocket. The patient was successfully treated with repositioning of the pump into the infraclavicular fossa.

LESSONS

Infraclavicular placement of the implanted pump allowed for a more secure pocket base for this patient and less strain applied to the pump, minimizing the risk of disruption of pump positioning and interruption of drug delivery.

ABBREVIATIONS

CP = cerebral palsy; ITB = intrathecal baclofen; GABA = γ-amino-butyric-acid

Intrathecal baclofen (ITB) therapy has been used to manage spasticity since the mid-1980s,1 and its use in children with cerebral palsy (CP) has increased over the past 15 years. Baclofen is a γ-amino-butyric-acid (GABA) agonist, and when administered to the spinal cord decreases spinal reflex activity and the number and severity of muscle spasms.2,3 Intrathecal administration of baclofen through an implanted catheter and pump (i.e., ITB) allows for a high concentration of baclofen to be delivered to the spinal cord. Compared to oral baclofen, ITB allows for smaller therapeutic doses, which eliminates most side effects related to systemic absorption while achieving improved clinical response.4,5 Numerous studies have reported the efficacy of ITB therapy in decreasing abnormal muscle tone and improving function, comfort, and ease of caregiving.2,6–10

The pump is most often placed in the subcutaneous or subfascial compartment of the lower abdomen to allow easy access for refills, facilitate tunneling to the spinal incision, and minimize the risk of skin breakdown or pressure injury. It is rare to encounter circumstances that dissuade abdominal placement of the device, and there are few such cases documented in the literature that describe alternative sites for pump placement.11–14 Here we describe a patient with recurrent displacement of their abdominal ITB pump leading to catheter malfunctions who necessitated repositioning of the pump to an alternate site, and review the relevant literature in order to assist future practitioners faced with this unusual clinical scenario.

Illustrative Case

A 17-year-old female, born at 28 weeks’ gestation with grade 4 intraventricular hemorrhage, posthemorrhagic hydrocephalus treated with a left-sided ventriculoperitoneal shunt, elevated body mass index (29 kg/m2), and epilepsy with spastic diplegia Gross Motor Function Classification level 4. She is verbal but with significant echolalia, and her self-reports are generally accurate. Mother and father are very involved and excellent historians.

As part of multimodality tone management, she underwent placement of an ITB pump (40 mL; Synchromed II, Medtronic Inc.) at age 12 in the subcutaneous space above the fascia in the right abdomen along with an Ascenda (Medtronic Inc.) catheter introduced through the lumbar spine with tip positioned at the T3 level. Clinical response was excellent with no issues for the first 3 years. Subsequently she began experiencing increased leg pains, tone, and agitation. Upon investigation, no motor stalls were noted. The volume of drug in the pump reservoir (40 mL) was higher than expected (19 mL), and drug/cerebrospinal fluid could not be aspirated from the side port, suggesting catheter occlusion. She was taken to the operating room and a tight bend of the catheter was discovered in the abdominal pocket, resulting in catheter occlusion. That section of tubing was replaced along with the pump, and the pump was secured with one Prolene stitch through the pump suture ring anchor, which is our routine for pump replacements in a mature pocket. An abdominal binder was used for 6 weeks postoperatively to help keep the pump secure during healing. Her ITB system functioned well for the next 12 months, although of note she underwent exploratory laparoscopy with an upper midline abdominal incision along with resection of a hepatic adenoma and cholecystectomy, a procedure during which the pump region was retracted temporarily.

Approximately a year after her pump/catheter revision surgery, she was again found to have a full reservoir when 13 mL were expected. The family thought the pump was lower in the abdomen and had turned, although radiographs showed no concerns with pump placement. Upon surgical exploration of the pump pocket, the pump was found to be floating freely. The catheter was tightly coiled from likely multiple pump rotations, causing occlusion proximal to the straight connector between the pump and spinal segments of catheter (Fig. 1). This coiled segment of tubing was resected along with replacement of the pump segment of catheter and the pump was secured with 2 Prolene sutures 2.0 to 2 different suture ring anchors. An abdominal binder was again used postoperatively to help keep the pump secure during healing.

FIG. 1.
FIG. 1.

Catheter is noted to be tightly coiled with a kink in the tubing.

Five months later she was noted to have episodes of increased tone and the family noted occasional unusual protrusion of the pump. She continued to wear an abdominal binder since the prior surgery to help keep the pump secure; however, radiographs showed the pump had flipped and rotated. The family suspected her favored crouched position, especially when sitting on the commode, was causing her iliac bone and ribs to slip behind the pump, and with the added stress of straining with a bowel movement, this could be causing shear stress on the pump and the repeated detachment from the fascia. The patient and family were convinced that she was not forcefully manipulating the pump. She again underwent surgical exploration and only 1 anchor suture remained attached to fascia with the pump very mobile and able to rotate in the pocket. The catheter was again crimping, likely related to twisting, which could have caused intermittent catheter occlusion and baclofen withdrawal. To attempt to prevent stress on the pump, a new epifascial pocket was created medial and inferior to the prior pocket such that it sat farther from the ribs and iliac bone. The pocket was kept as small as possible, and the pump was secured to the fascia with 2.0 Prolene sutures through all 4 available anchor sites (our practice is normally to use 2 anchor sites on initial placement). A binder was again used postoperatively.

Less than 2 months later, the family was already concerned it may be protruding more again. Approximately 4 months after surgery, increased tone was noted, and radiographs again revealed a flipped pump.

After a long discussion with the patient and family, they strongly wished to continue receiving therapy rather than opt for device explantation. Weaning the intrathecal baclofen dose resulted in worsening symptoms, confirming it was still providing intermittent benefit. We decided to investigate alternative locations for the pump and after an exhaustive review of the literature (Table 1) and discussion with Medtronic regarding nonstandard locations, the right infraclavicular area was preferred. This would place it away from the patient’s left-sided ventriculoperitoneal shunt and allow us to secure the pump down to the pectoralis fascia. Given that this was a relatively stationary part of the body compared to the abdomen, it would eliminate the element of flexion, compression and dislodgment of the pump by the rib cage on the pelvis that we felt had been occurring previously. The patient had sufficient soft tissue overlying this region to accommodate the pump without significant surface protrusion.

TABLE 1.

Review of the literature

Authors & YearNo. of PatientsAlternative Pump Site
Rocque & Albright, 2010144Infraclavicular
Patel et al., 2021121Subpectoral
Narang et al., 2016119Upper thigh
Devine et al., 2016132Medial thigh & iliac fossa

At surgery, we found only 1 suture still attached to fascia and the pump able to freely rotate in the pocket. The catheter was coiled multiple times (Fig. 2). A 20-mL size pump was secured in a new subcutaneous pocket above the pectoralis fascia through an infraclavicular incision, and a new pump segment of catheter was tunneled from the abdomen to the chest. Postoperative care included the use of a sports bra or post-breast reduction bra to support the pump and prevent pocket expansion as it healed.

FIG. 2.
FIG. 2.

Catheter with significant coiling indicating excessive flipping of the pump in the pocket.

Sixteen months after surgery, the patient continues to do well with no concerns for pump malfunction or flipping. She reports there had been some discomfort in the abdominal site during the time she had the pump implanted there but now reports no pain in either site. She and her family are very happy with the new placement of the pump and no concerns have been raised related to the location. Refilling the pump in clinic is a bit more challenging in the new location due to excessive breast tissue. We have found refill in the sitting position to be most successful.

Discussion

Observations

Positioning an intrathecal pump in a nonabdominal location is rarely needed or reported. Our search revealed alternative locations published previously by 4 other authors (Table 1). One report did review 4 cases of infraclavicular pump placement, with one involving repositioning of an abdominal pump but also replacing the original lumbar catheter with a cervical spine inserted catheter. In our case, the lumbar catheter was preserved by tunneling the pump segment from the abdomen to the infraclavicular pocket, avoiding the need to place a cervical catheter. When planning infraclavicular placement of an intrathecal pump, there are some considerations to be made. First, patient/family acknowledgment that this location will likely be more visible than the abdomen. In our case, the patient has moderate breast tissue and the 20-mL pump is actually not visible. Second, specific placement of the pump in relation to the breast tissue (if present) should be considered, especially in anticipation of refill procedures. We found that the location could have been a little bit more superior (1–2 finger breadths below the clavicle) to minimize interference of breast tissue during the refill procedure. We also considered subfacial pump placement but believed that refills would have been prohibitively difficult due to soft tissue thickness and pump depth.

Lessons

When reviewing the potential causes of the recurrent displacement of the abdominal pump, we wonder if the laparoscopic abdominal surgery between the first and second pump surgical events contributed to the vulnerability of the pump to flip. The patient’s elevated body mass index and body posture could also have been risk factors that interfered with the security of the pump in the abdomen. In our decision-making process, we did also consider buttock, flank, and thigh placement of the pump for this patient and determined these to be less desirable options due to the patient’s size, wheelchair dependence and preference.

In conclusion, infraclavicular baclofen pump placement with tunneling to a lumbar spinal catheter for this patient has proven to be a success after more than 1 year and could be considered in similar cases, in both the pediatric and adult populations.

Disclosures

The authors report no conflict of interest concerning the materials or methods used in this study or the findings specified in this paper.

Author Contributions

Conception and design: Buxton, Morgan, Stone. Acquisition of data: Stone. Analysis and interpretation of data: Northam, Stone. Drafting the article: Buxton, Morgan, Stone. Critically revising the article: Buxton, Northam, Stone. Reviewed submitted version of manuscript: all authors. Approved the final version of the manuscript on behalf of all authors: Buxton. Administrative/technical/material support: Stone. Study supervision: Stone.

References

  • 1

    Penn RD, Kroin JS. Continuous intrathecal baclofen for severe spasticity. Lancet. 1985;2(8447):125127.

  • 2

    Armstrong RW, Steinbok P, Cochrane DD, Kube SD, Fife SE, Farrell K. Intrathecally administered baclofen for treatment of children with spasticity of cerebral origin. J Neurosurg. 1997;87(3):409414.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 3

    Nuttin B, Ivanhoe C, Albright L, Dimitrijevic M, Saltuari L. Intrathecal baclofen therapy for spasticity of cerebral origin: cerebral palsy and brain injury. Neuromodulation. 1999;2(2):120132.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 4

    Albright AL. Intrathecal baclofen in cerebral palsy movement disorders. J Child Neurol. 1996;11(suppl 1):S29S35.

  • 5

    Gilmartin R, Bruce D, Storrs BB, et al. Intrathecal baclofen for management of spastic cerebral palsy: multicenter trial. J Child Neurol. 2000;15(2):7177.

  • 6

    Gerszten PC, Albright AL, Johnstone GF. Intrathecal baclofen infusion and subsequent orthopedic surgery in patients with spastic cerebral palsy. J Neurosurg. 1998;88(6):10091013.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 7

    Albright AL, Barron WB, Fasick MP, Polinko P, Janosky J. Continuous intrathecal baclofen infusion for spasticity of cerebral origin. JAMA. 1993;270(20):24752477.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 8

    Awaad Y, Tayem H, Munoz S, Ham S, Michon AM, Awaad R. Functional assessment following intrathecal baclofen therapy in children with spastic cerebral palsy. J Child Neurol. 2003;18(1):2634.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 9

    Campbell SK, Almeida GL, Penn RD, Corcos DM. The effects of intrathecally administered baclofen on function in patients with spasticity. Phys Ther. 1995;75(5):352362.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 10

    Gooch JL, Oberg WA, Grams B, Ward LA, Walker ML. Care provider assessment of intrathecal baclofen in children. Dev Med Child Neurol. 2004;46(8):548552.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 11

    Narang S, Srinivasan SK, Zinboonyahgoon N, Sampson CE. Upper antero-medial thigh as an alternative site for implantation of intrathecal pumps: a case series. Neuromodulation. 2016;19(6):655663.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 12

    Patel AA, Patel CB, Shah NA, Jassal NS, Padalia DM. Alternative implantation site of intrathecal pump in the submuscular plane of the pectoralis major muscle: a case report. A A Pract. 2021;15(6):e01464.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 13

    Devine O, Harborne A, Lo WB, Weinberg D, Ciras M, Price R. Unusual placement of intrathecal baclofen pumps: report of two cases. Acta Neurochir (Wien). 2016;158(1):167170.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 14

    Rocque BG, Albright AL. Infraclavicular fossa as an alternate site for placement of intrathecal infusion pumps: technical note. Neurosurgery. 2010;66(2):E402E403.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • Collapse
  • Expand
  • FIG. 1.

    Catheter is noted to be tightly coiled with a kink in the tubing.

  • FIG. 2.

    Catheter with significant coiling indicating excessive flipping of the pump in the pocket.

  • 1

    Penn RD, Kroin JS. Continuous intrathecal baclofen for severe spasticity. Lancet. 1985;2(8447):125127.

  • 2

    Armstrong RW, Steinbok P, Cochrane DD, Kube SD, Fife SE, Farrell K. Intrathecally administered baclofen for treatment of children with spasticity of cerebral origin. J Neurosurg. 1997;87(3):409414.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 3

    Nuttin B, Ivanhoe C, Albright L, Dimitrijevic M, Saltuari L. Intrathecal baclofen therapy for spasticity of cerebral origin: cerebral palsy and brain injury. Neuromodulation. 1999;2(2):120132.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 4

    Albright AL. Intrathecal baclofen in cerebral palsy movement disorders. J Child Neurol. 1996;11(suppl 1):S29S35.

  • 5

    Gilmartin R, Bruce D, Storrs BB, et al. Intrathecal baclofen for management of spastic cerebral palsy: multicenter trial. J Child Neurol. 2000;15(2):7177.

  • 6

    Gerszten PC, Albright AL, Johnstone GF. Intrathecal baclofen infusion and subsequent orthopedic surgery in patients with spastic cerebral palsy. J Neurosurg. 1998;88(6):10091013.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 7

    Albright AL, Barron WB, Fasick MP, Polinko P, Janosky J. Continuous intrathecal baclofen infusion for spasticity of cerebral origin. JAMA. 1993;270(20):24752477.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 8

    Awaad Y, Tayem H, Munoz S, Ham S, Michon AM, Awaad R. Functional assessment following intrathecal baclofen therapy in children with spastic cerebral palsy. J Child Neurol. 2003;18(1):2634.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 9

    Campbell SK, Almeida GL, Penn RD, Corcos DM. The effects of intrathecally administered baclofen on function in patients with spasticity. Phys Ther. 1995;75(5):352362.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 10

    Gooch JL, Oberg WA, Grams B, Ward LA, Walker ML. Care provider assessment of intrathecal baclofen in children. Dev Med Child Neurol. 2004;46(8):548552.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 11

    Narang S, Srinivasan SK, Zinboonyahgoon N, Sampson CE. Upper antero-medial thigh as an alternative site for implantation of intrathecal pumps: a case series. Neuromodulation. 2016;19(6):655663.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 12

    Patel AA, Patel CB, Shah NA, Jassal NS, Padalia DM. Alternative implantation site of intrathecal pump in the submuscular plane of the pectoralis major muscle: a case report. A A Pract. 2021;15(6):e01464.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 13

    Devine O, Harborne A, Lo WB, Weinberg D, Ciras M, Price R. Unusual placement of intrathecal baclofen pumps: report of two cases. Acta Neurochir (Wien). 2016;158(1):167170.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 14

    Rocque BG, Albright AL. Infraclavicular fossa as an alternate site for placement of intrathecal infusion pumps: technical note. Neurosurgery. 2010;66(2):E402E403.

    • PubMed
    • Search Google Scholar
    • Export Citation

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