ORIGINAL ARTICLE

A Wearable Epidural Catheter Infusion System for Patients With Intractable Spontaneous Intracranial Hypotension Wouter I. Schievink, MD,* Howard L. Rosner, MD,† and Charles Louy, MD, PhD† Background and Objectives: Spontaneous intracranial hypotension is an important cause of secondary headaches, and most patients respond well to epidural blood patching or direct repair of the underlying spinal cerebrospinal fluid leak. However, options are limited for those patients who have exhausted these traditional treatments, especially when spinal imaging is normal. We describe a wearable epidural catheter infusion system for patients with intractable spontaneous intracranial hypotension. Methods: Six patients with intractable spontaneous intracranial hypotension (4 women and 2 men; mean age, 53 years; mean duration of symptoms, 50 months) underwent placement of a permanent indwelling spinal epidural catheter attached to an external infusion pump. The Migraine Disability Assessment questionnaire was used to assess the severity of the symptoms, before and during treatment. Results: The infusion resulted in complete or near-complete symptom relief in 5 of 6 patients (Migraine Disability Assessment score decreased from grade IV to grade I or II). However, the epidural catheter infusion system was removed in 2 patients because of infection, in 1 patient because of delayed failure to provide adequate symptom control, and in 1 patient because of minimal symptom relief. Two patients reported excellent and sustained symptom relief over 27 and 36 months of follow-up. Conclusions: This wearable epidural catheter infusion system showed promising efficacy results but the high rate of complications limits its use to a very select group of patients. (Reg Anesth Pain Med 2015;40: 49–51)

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pontaneous intracranial hypotension is an important cause of secondary headaches, often afflicting otherwise healthy young and middle-aged adults.1 The cause of spontaneous intracranial hypotension is almost always a spontaneous cerebrospinal fluid (CSF) leak at the level of the spine. Fortunately, most of the patients respond well to a trial of bed rest or the placement of one or more epidural blood patches.1 Injections of fibrin glue or surgical repair of the underlying spinal CSF leak are often effective in those in whom less invasive treatments have failed. However, options are limited for those patients with recalcitrant symptoms of spontaneous intracranial hypotension who have exhausted these traditional treatments, especially when spinal imaging is not able to localize the level of the CSF leak.1,2 Epidural saline infusions generally provide excellent relief of symptoms of spontaneous intracranial hypotension,3–5 but in our experience only for the duration of the infusion. The proposed mechanism of action of epidural saline infusion is that of producing splinting pressures in the epidural space. Here, we report a trial of a wearable epidural

From the Departments of *Neurosurgery and †Anesthesiology, Cedars-Sinai Medical Center, Los Angeles, CA. Accepted for publication October 20, 2014. Address correspondence to: Wouter I. Schievink, MD, Department of Neurosurgery, Cedars-Sinai Medical Center, 127 S San Vicente Blvd, Ste A6600, Los Angeles, CA 90048 (e‐mail: [email protected]). The authors declare no conflict of interest. Copyright © 2014 by American Society of Regional Anesthesia and Pain Medicine ISSN: 1098-7339 DOI: 10.1097/AAP.0000000000000192

catheter infusion system for patients with intractable spontaneous intracranial hypotension.

METHODS Patients Six patients with spontaneous intracranial hypotension were recruited for this trial (Table 1). The insertions of the spinal epidural catheters were performed between June 2009 and January 2010. Four of the patients were women and the mean age of the group was 53 years (range, 40–79 years). Duration of symptoms varied from 24 to 105 months (mean, 50 months). Orthostatic headache was the most prominent complaint in all patients. Neurologic examination and brain magnetic resonance imaging (MRI) findings were normal in all patients. Initial opening pressure at time of lumbar puncture was below normal in all patients (range, negative to 5 cm H2O). Spinal MRI and computed tomography-myelography was performed in all patients and showed an extensive CSF leak in 1 patient and multiple spinal meningeal diverticula in 2 patients. Spinal imaging was entirely normal in the remaining 3 patients. Thus, the presence of a spinal CSF leak could not be established in 5 of the 6 patients. All patients had undergone 2 or more epidural blood patches. Three patients had undergone 1 or more surgical treatments directed at the CSF leak or largest spinal meningeal diverticula. All patients continued with debilitating headaches despite these treatments, although transient benefit was reported by all patients after these treatments. This study was approved by our medical center’s institutional review board. All patients consented for their de-identified information to be included. All patients completed a Migraine Disability Assessment (MiDAS) questionnaire to assess the severity of the symptoms, before and during treatment.6 A MiDAS score of 0 to 5 (grade I) is considered to equate to little or no disability, 6 to10 (grade II) is mild disability, 11 to 20 (grade III) is moderate disability, and more than 20 (grade IV) is severe disability.6

Procedures Initially, a trial of epidural saline infusion lasting between 48 and 72 hours was performed. An epidural catheter was placed under fluoroscopy at the L2-3 or L3-4 level and the catheter was attached to an infusion pump. Sterile preservative-free normal saline or a proprietary buffered saline solution (Elliot’s B solution; QOL Medical, Vero Beach, Florida) was used as the infusate. Patients were hospitalized and encouraged to ambulate and be in the upright position to assess effectiveness of the infusion. All patients reported excellent headache relief at infusion rates between 4 and 12.5 mL/h. The spinal epidural catheters were then removed and, the next day, the permanent indwelling epidural catheter infusion system was placed. Under general endotracheal anesthesia, the patient was placed in the prone or lateral decubitus position. Using a 16-gauge Tuohy needle, the epidural space was entered at the L2-3 or L3-4 level and an epidural spinal catheter (Du-Pen catheter; Bard Access Systems, Salt Lake City, Utah, or Portex catheter, Smiths-Medical, St Paul,

Regional Anesthesia and Pain Medicine • Volume 40, Number 1, January-February 2015

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Regional Anesthesia and Pain Medicine • Volume 40, Number 1, January-February 2015

Schievink et al

TABLE 1. Clinical and Radiographic Data on 6 Patients Who Underwent Placement of a Permanent Indwelling Epidural Catheter Infusion System

Age, y/Sex

Duration of Symptoms, mo

Opening Pressure, cm H2O

40/F

37

2

48/F

41

79/M

Imaging

Previous Treatments No. CT Myelograms

No. EBPs

No. Surgeries

Outcome of Epidural Infusion System

Normal Meningeal diverticula

4

3 lumbar and thoracic

1

5

Normal Normal

2

6 lumbar only

0

59

2

Normal Normal

3

6 lumbar only

0

48/F

105

Negative

9

33

0

16 lumbar and thoracic 2 lumbar only

4

48/M

Normal Cervicothoracic CSF leak Normal Normal

56/F

24

4

Normal Meningeal diverticula

6

Excellent symptom relief for 5 mo, then developed infection Excellent symptom relief for 27 mo, then spontaneous resolution of spontaneous intracranial hypotension Minimal symptom relief for 7 mo, then removal of device Excellent symptom relief for 5 mo, then lost effectiveness Excellent sustained symptom relief during 36 mo of follow-up Excellent symptom relief for 3 wk, then developed infection

Brain

Spine

2

8 lumbar and thoracic

0 3

CT indicates computed tomography, EBPs, epidural blood patches.

Minnesota) was threaded in the posterior epidural space to the T9 or T10 level using fluoroscopic guidance. Epidurograms were performed to confirm localization. The catheter was then tunneled to the anterior chest wall where it was attached to an infusion port overlying a rib (Fig. 1). A single dose of intravenous antibiotics was given just before the start of the procedure.

RESULTS All 6 patients lived outside of California and follow-up care was provided by their local primary care provider, neurologist, and neurosurgeon. There were no immediate operative complications. Using a Huber needle, the port was first accessed within 6 hours to 1 week postoperatively. The needle was connected to the wearable infusion system, consisting of an infusion pump which can be carried in a belt pack. Only sterile preservativefree normal saline was used as the infusate. Patients were advised to adjust infusion volumes and rates to optimize effectiveness and comfort. Eventually, mean infusion rates of 6 to 15 mL/h were used for 10 to 18 hours/d. The needle was exchanged between once daily to once every 7 to 10 days. Needle insertions were performed at a physician’s office in 2 patients, at home by a home health nurse in 1 patient, and at home by the patient (or spouse) in 3 patients. The infusion resulted in complete or near-complete symptom relief in 5 of 6 patients. The MiDAS score decreased from grade IV (severe disability) to grade I or II (none to mild disability) in these 5 patients. An infection developed in 2 patients, after 3 weeks and 5 months, respectively, necessitating removal of the system. The first patient presented with worsening headache, fever (39.2°F), nuchal rigidity, and erythema of the incision sites. A CSF examination showed the following values: white blood cell count, 1395 microliter; total protein, 1706 mg/dL; and glucose, 6 mg/dL. Intravenous antibiotics were started, the systems were removed, and the patient made an uneventful recovery. Cultures showed pseudomonas. The second patient noted erythema at the port site and

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then along the catheter track. The system was removed and the patient made an uneventful recovery. Cultures showed clostridium and she was treated with a 10-day course of antibiotics. These patients continue to be symptomatic with orthostatic headaches. Of note, these patients who developed an infection exchanged the needle once every 1 to 2 days and the 4 patients who did not develop an infection exchanged the needle every 5 to 10 days. In 1 patient, infusions became more difficult over time and effectiveness was greatly reduced, possibly related to epidural scar formation. The system was removed at 5 months, postoperatively. This patient continues to be symptomatic with orthostatic headaches. In 1 patient, the epidural infusions only provided minimal symptom relief (MiDAS score remained at grade IV). The system was removed at 7 months postoperatively. This patient continues to be symptomatic with orthostatic headaches. The remaining 2 patients enjoyed long-term sustained effective headache relief. One patient noted the development of a highpressure headache at month 27 postoperatively that progressed during a 2-week period. The infusion was discontinued and the patient has remained asymptomatic for an additional 12-month follow-up period. We postulate that the spontaneous intracranial hypotension resolved spontaneously in this patient. Alternatively, the prolonged epidural infusion could have contributed directly to resolution of the intracranial hypotension. The other patient continues with the infusions with excellent headache relief at last follow-up 36 months postoperatively, although replacement of the catheter was required at 30 months postoperatively due to catheter fracture.

DISCUSSION In this report, we describe a novel surgical technique for the treatment of patients with intractable headache due to spontaneous intracranial hypotension. The objective was to provide headache relief with minimal interference with the patients’ activities of © 2014 American Society of Regional Anesthesia and Pain Medicine

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Regional Anesthesia and Pain Medicine • Volume 40, Number 1, January-February 2015

Implantable Epidural Catheter for SIH

FIGURE 1. A, In the lateral decubitus position, a Tuohy needle is placed in the lumbar epidural space. B, An epidurogram is performed to confirm localization. C, The catheter is threaded into the epidural space. D, Fluoroscopy is performed to confirm localization. E, The port-acath is shown. F, Using a short shunt passer, a subcutaneous tunnel is fashioned between the lumbar and chest incisions. G, Loops of catheter are placed subcutaneously in the lumbar pocket. H, Finally, the port-a-cath is placed in the subcutaneous pocket along with loops of catheter.

daily living. Indeed, as predicted by the positive response to the trial epidural saline infusion, the continuous epidural infusions provided excellent pain relief in all but 1 patient. The patients were able to access the port with minimal inconvenience and the wearable nature of the infusion pump provided good portability. However, there were a significant number of complications— necessitating removal of the epidural infusion system in 4 of the 6 patients. Complications included infections, catheter fracture, and delayed failure to provide adequate symptom control. Two of the 6 patients experienced long-term sustained effectiveness. Most, but not all, patients with spontaneous intracranial hypotension have characteristic findings on brain MRI, such as brain sagging, pachymeningeal enhancement, and subdural fluid collections. It has been shown that patients with spontaneous intracranial hypotension and a normal brain MRI are more difficult to treat7 and—not surprisingly—all of the 6 patients in the present study had a normal brain MRI. To obtain adequate pain relief, relatively high volumes between 4 and 12.5 mL/h were required for epidural spinal infusion and patients used the device for most hours of the day. This precludes the use of fully implantable pumps which have a maximal capacity of approximately 20 mL. The external pump used in this study was associated with a significant infection risk and it was notable that those patients who developed an infection had accessed the port on a daily basis and those who did not develop an infection had accessed the port on a weekly basis. Despite the difficulties encountered by our patients, placement of a permanent indwelling epidural catheter infusion system has a place—albeit very limited—in the armamentarium of physicians treating patients with recalcitrant spontaneous intracranial © 2014 American Society of Regional Anesthesia and Pain Medicine

hypotension. Because of the high early failure rate, we had discontinued the use of this procedure, but because of the sustained effectiveness in some of the patients and upon their encouragement, we have again offered this procedure to selected patients who have debilitating symptoms and have exhausted all other reasonable options.

REFERENCES 1. Schievink WI. Spontaneous spinal cerebrospinal fluid leaks and intracranial hypotension. JAMA. 2006;295:2286–2296. 2. Schievink WI. A novel technique for treatment of intractable spontaneous intracranial hypotension: lumbar dural reduction surgery. Headache. 2009; 49:1047–1051. 3. Gibson BE, Wedel DJ, Faust RJ, Petersen RC. Continuous epidural saline infusion for the treatment of low CSF pressure headache. Anesthesiology. 1988;68:789–791. 4. Kawasaki S, Yamamoto Y, Sunami N, Suga M, Mizumatsu S, Inoue T. Treatment of spontaneous intracranial hypotension with continuous epidural saline infusion: a case report [in Japanese]. No To Shinkei. 1999;51:711–715. 5. Rouaud T, Lallement F, Choui R, Madigand M. Treatment of spontaneous intracranial hypotension by epidural saline infusion. Rev Neurol. 2009;165: 201–205. 6. Stewart WF, Lipton RB, Whyte J, et al. An international study to assess reliability of the Migraine Disability Assessment (MIDAS) score. Neurology. 1999;53:988–994. 7. Schievink WI, Maya MM, Louy C. Cranial MRI predicts outcome of spontaneous intracranial hypotension. Neurology. 2005;64:1282–1284.

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Copyright © 2014 American Society of Regional Anesthesia and Pain Medicine. Unauthorized reproduction of this article is prohibited.

A wearable epidural catheter infusion system for patients with intractable spontaneous intracranial hypotension.

Spontaneous intracranial hypotension is an important cause of secondary headaches, and most patients respond well to epidural blood patching or direct...
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