J Neurosurg Pediatrics 12:357–359, 2013 ©AANS, 2013

Medical management of eosinophilic meningitis following bovine graft duraplasty for Chiari malformation Type I repair Case report Adam P. Ostendorf, M.D.,1 and Anne M. Connolly, M.D.1,2 Departments of 1Neurology and 2Pediatrics, Washington University School of Medicine, St. Louis, Missouri Eosinophilic meningitis is a known complication of duraplasty, including that using bovine tissues. Previous authors have relied on surgical removal of the graft for treatment. Authors of the present report describe a 7-year-old girl with eosinophilic meningitis following duraplasty with a bovine pericardium graft who was successfully treated using corticosteroid therapy alone. (http://thejns.org/doi/abs/10.3171/2013.7.PEDS13130)

Key Words      •      eosinophilic meningitis      •      pediatric neurosurgery      •      duraplasty      • bovine graft      •      corticosteroid      •      Chiari malformation

E

osinophilic meningitis is most often associated with

helminthic infection but has been noted to occur in reaction to other infectious processes or neoplastic disease as well as in the setting of foreign bodies within the CNS.8 One report of eosinophilic meningitis following duraplasty with a bovine graft has been published, and the patient in that case fully recovered following surgical removal of the graft.7 Another case series included a patient with lymphocytic meningitis without eosinophilic graft infiltration following a duraplasty utilizing the same type of bovine graft in combination with DuraSeal (Covidien).14 Here, we describe a patient with eosinophilic meningitis following Chiari decompression via bovine graft duraplasty with resolution following treatment with corticosteroids. Approval was provided through the Washington University Human Research Protection Office under the Guideline for Case Reports and Case Series.

Case Report History. A 7-year-old girl with a history of focal onset seizures, headaches, and gross motor delay underwent Chiari malformation Type I decompression with suboccipital craniotomy, C-1 laminectomy, and duraplasty at an outside institution. The duraplasty was performed using Abbreviations used in this paper: CBC = complete blood count; WBC = white blood cell.

J Neurosurg: Pediatrics / Volume 12 / October 2013

Dura-Guard (Synovis Surgical Innovations) and a bovine pericardium-based graft; closure was achieved using DuraSeal, a polyethylene glycol hydrogel sealant. The patient lived on a farm with two dogs, had recently traveled to the East Coast, swam in Missouri rivers and ponds, and camped. She had no history of ingesting undercooked foods. She was taking carbamazepine and levetiracetam for epilepsy and did not use ibuprofen prior to the onset of symptoms. Approximately 2 weeks following her surgery, she exhibited irritability and fatigue, followed by nausea, vomiting, low-grade fever, and headache over the next day. She was admitted to the referring hospital and treated with antiemetics, which led to improvement in the nausea and vomiting. Early in her hospital course, she received ceftriaxone and vancomycin for 24 hours following the growth of Staphylococcus epidermidis in 1 of 2 blood culture bottles. She continued to have a nonpositional, moderately severe headache over the vertex that was associated with photophobia and mild posterior neck pain. Five days after admission, with persistent headache, low-grade fever, irritability, and neck pain, she underwent lumbar puncture and brain MRI. The lumbar puncture revealed an opening pressure of 27 cm H2O in the setting of propofol and ketamine sedation. She had 40 red blood cells/ml and 590 leukocytes/ml with 63% eosinophils and 35% lymphocytes, and her protein level was 75 mg/dl with a glucose level of 20 mg/dl. Her CBC revealed 6000 WBCs/ml with 6% eosinophils. 357

A. P. Ostendorf and A. M. Connolly Brain and spine MRI revealed postoperative changes, including a collection at the suboccipital craniotomy site without rim enhancement (Fig. 1). She was subsequently transferred to our hospital for further evaluation and treatment.

Examination. On our initial examination, 3 weeks af­ter her surgery, she appeared irritable and in moderate pain. She had tenderness to palpation of her surgical site but no surrounding erythema, induration, fluctuance, or palpable fluid collections. Her neck range of motion was normal without Brudzinski or Kernig signs. She had mild photosensitivity. She also had a broad-based gait that was slightly unsteady, and she could not perform tandem gait. Repeat CSF analysis following admission revealed 641 nucleated cells/ml with 25% eosinophils and 68% lymphocytes. Her CSF glucose level was < 20 mg/dl despite a serum glucose level over 90 mg/dl, and her CSF protein level was 62 mg/dl. A CBC demonstrated 6700 WBCs/ml with 2.7% eosinophils. Cytopathology showed no malignant cells. An infectious disease evaluation of the blood, CSF, and stool was negative, including ova and parasite examination; bacteria, mycobacteria, and fungal cultures; and serological evaluations for antibodies to toxoplasma, Toxocara, HIV, Paragonimus, Enterovirus, and Cryptococcus. Mantoux skin testing was also negative.

Treatment. She was initially placed on cefepime, vancomycin, and doxycycline in addition to her home medications of levetiracetam and carbamazepine. She was started on intravenous dexamethasone 2 mg every 6 hours, which was 0.25 mg/kg/day, with subsequent improvement in her neurological symptoms over the following 3 days.

Posttreatment Course. At discharge, her neurological examination was at her baseline, which was normal except for an inability to perform tandem gait. Her symptoms returned after her steroid dose was decreased on Day 5 of treatment. The symptoms immediately resolved after increasing her dose to 2 mg every 6 hours and continuing this dose for 4 weeks, followed by a 2-week taper. After completing a total of 5 weeks of high-dose corticosteroids with a 2-week taper, she was symptom free and remained so at her 6-month follow-up appointment.

Discussion

Eosinophilic meningitis is most often associated with helminthic infection but has been described in neoplastic disease, in reaction to drugs or dyes, and after implantation of plastic devices.9 Aseptic meningitis has been reported following duraplasty with a wide variety of graft materials, including cadaveric and synthetic grafts.1,2,6,9,12,14 Recently, aseptic meningitis developed in three individuals after duraplasty with Lyoplant (Aesculap Inc.), a bovine pericardium collagen implant; all three were successfully treated using steroids, although the details of the cellular infiltrate and treatment were not reported.10 Several studies have documented outcomes following duraplasty using bovine pericardium without mention of eosinophilic meningitis.3–5,13,17 One occurrence of eosinophilic meningitis following duraplasty with Dura-Guard 358

Fig. 1.  Postoperative sagittal T1-weighted (left) and axial T1-weighted post–Gd administration (right) MR images of the brain and cervical spinal cord revealing cerebellar tonsillar ectopia descending to the level of the C-2 ossification center with postoperative CSF collection (asterisk) at the suboccipital craniotomy site. There is no associated contrast enhancement at the level of the fluid collection. The axial image was captured at the level of the white line in the left image.

was reported to the US Food and Drug Administration, although the treatment and outcome were not detailed.16 Eosinophilic meningitis has been reported following duraplasty with other bovine tissue derivatives. A case report described eosinophilic meningitis and graft infiltration following the use of Durepair (TEI Biosciences), a graft derived from fetal bovine skin.7 In addition, a case series of complications following Chiari decompression in children documented lymphocytic meningitis without eosinophilic graft infiltration in one child following duraplasty with Durepair and DuraSeal.14 These two patients were treated with the removal of the bovine graft, and symptoms subsequently resolved. The patient in our case likely developed eosinophilic meningitis in reaction to the bovine pericardium dural graft. She had no evidence of infectious causes or neoplastic disease despite an extensive evaluation. Furthermore, she was not taking a medication previously associated with eosinophilic meningitis. We report the first documented case of eosinophilic meningitis in the setting of bovine tissue dural graft with resolution following medical management. Two other pa­tients were successfully treated only after surgical replacement of the dural graft.7,14 Three other patients with aseptic meningitis following duraplasty with Lyoplant re­sponded to corticosteroids, although it is unknown if they had eosinophilic meningitis and details regarding the doses and duration of treatment were not reported.10 In both our patient and another previously described, symptoms relapsed after decreasing or ceasing cor­ticosteroids at 5–14 days of treatment.7 Utilizing a longer course of corticosteroids with tapering may prevent surgical revision. Perhaps a longer course of immunosuppression provides time for host tissue to replace the graft. Besides differences in the duration and dosing of corticosteroids, another possible cause for an alternative clinical course is the difference between the immunogenicity of the grafts. Dura-Guard is bovine pericardium that is manufactured with a glutaraldehydecollagen cross-linking process, which is thought to lead J Neurosurg: Pediatrics / Volume 12 / October 2013

Eosinophilic reaction to bovine dural graft treated with steroids to lower immunogenicity,15 whereas Durepair is derived from fetal bovine skin and fibrin glue and the collagen fibrils are not cross-linked.18 Lyoplant is manufactured with a controlled lyophilization process and has been shown to activate a T-helper cell Type 2 immunogenic response, suppress eosinophil cytokine release, and produce a more lymphocytic response.11

Conclusions

To summarize, we described the successful medical management of eosinophilic meningitis following bovine graft duraplasty. Previous cases have been treated via surgical revision with removal of the graft. We recommend a trial of at least 4 weeks of high-dose corticosteroids with a prolonged tapering over at least 2 weeks as an alternative approach to surgical removal of the graft. This approach may provide results similar to, and lower morbidity than, those with surgical management. Disclosure 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 to the study and manuscript preparation include the following. Conception and design: Ostendorf. Analysis and interpretation of data: Ostendorf. Drafting the article: Ostendorf. Critically revising the article: both authors. Reviewed submitted version of manuscript: both authors. Approved the final version of the manuscript on behalf of both authors: Ostendorf. Study supervision: Connolly. References   1.  Abla AA, Link T, Fusco D, Wilson DA, Sonntag VK: Comparison of dural grafts in Chiari decompression surgery: review of the literature. J Craniovertebr Junction Spine 1:29–37, 2010   2.  Alleyne CH Jr, Barrow DL: Immune response in hosts with cadaveric dural grafts. Report of two cases. J Neurosurg 81: 610–613, 1994   3.  Anson JA, Marchand EP: Bovine pericardium for dural grafts: clinical results in 35 patients. Neurosurgery 39:764–768, 1996   4.  Baharuddin A, Go BT, Firdaus MN, Abdullah J: Bovine pericardium for dural graft: clinical results in 22 patients. Clin Neu­­rol Neurosurg 104:342–344, 2002   5.  Filippi R, Schwarz M, Voth D, Reisch R, Grunert P, Perneczky A: Bovine pericardium for duraplasty: clinical results in 32 patients. Neurosurg Rev 24:103–107, 2001   6.  Foreman P, Safavi-Abbasi S, Talley MC, Boeckman L, Mapstone TB: Perioperative outcomes and complications associated with allogeneic duraplasty for the management of Chiari malformations Type I in 48 pediatric patients. Clinical article. J Neurosurg Pediatr 10:142–149, 2012

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  7.  Foy AB, Giannini C, Raffel C: Allergic reaction to a bovine dural substitute following spinal cord untethering. Case report. J Neurosurg Pediatr 1:167–169, 2008   8.  Graeff-Teixeira C, da Silva AC, Yoshimura K: Update on eosinophilic meningoencephalitis and its clinical relevance. Clin Microbiol Rev 22:322–348, 2009   9.  Lam FC, Kasper E: Augmented autologous pericranium dura­ plasty in 100 posterior fossa surgeries—a retrospective case series. Neurosurgery 71 (2 Suppl Operative):ons302–ons307, 2012 10.  Litvack ZN, Lindsay RA, Selden NR: Dura splitting decompression for Chiari I malformation in pediatric patients: clinical outcomes, healthcare costs and resource utilization. Neurosurgery 72:922–929, 2013 11.  Meyer T, Meyer B, Schwarz K, Höcht B: Immune response to xenogeneic matrix grafts used in pediatric surgery. Eur J Pediatr Surg 17:420–425, 2007 12.  Munshi I, Frim D, Stine-Reyes R, Weir BK, Hekmatpanah J, Brown F: Effects of posterior fossa decompression with and without duraplasty on Chiari malformation-associated hydromyelia. Neurosurgery 46:1384–1390, 2000 13.  Parízek J, Mĕricka P, Spacek J, Nĕmecek S, Eliás P, Sercl M: Xenogeneic pericardium as a dural substitute in reconstruction of suboccipital dura mater in children. J Neurosurg 70:905– 909, 1989 14.  Parker SR, Harris P, Cummings TJ, George T, Fuchs H, Grant G: Complications following decompression of Chiari malformation Type I in children: dural graft or sealant? Clinical article. J Neurosurg Pediatr 8:177–183, 2011 15.  Speer DP, Chvapil M, Eskelson CD, Ulreich J: Biological effects of residual glutaraldehyde in glutaraldehyde-tanned collagen biomaterials. J Biomed Mater Res 14:753–764, 1980 16.  US Food and Drug Administration: MAUDE adverse event report: Bio-vascular, Inc. Dura-Guardduarl patch. (http:// www.accessdata.fda.gov/scripts/cdrh/cfdocs/cfMAUDE/ detail.cfm?mdrfoi__id=232872). [Accessed July 15, 2013] 17.  Williams LE, Vannemreddy PS, Watson KS, Slavin KV: The need in dural graft suturing in Chiari I malformation decompression: a prospective, single-blind, randomized trial comparing sutured and sutureless duraplasty materials. Surg Neurol Int 4:26, 2013 18.  Zerris VA, James KS, Roberts JB, Bell E, Heilman CB: Repair of the dura mater with processed collagen devices. J Biomed Mater Res B Appl Biomater 83:580–588, 2007

Manuscript submitted March 15, 2013. Accepted July 11, 2013. Please include this information when citing this paper: published online August 2, 2013; DOI: 10.3171/2013.7.PEDS13130. Address correspondence to: Adam P. Ostendorf, M.D., De­­ partment of Neurology, Washington University School of Medicine, Box 8111, 660 S. Euclid Ave., St. Louis, MO 63110. email: [email protected].

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Medical management of eosinophilic meningitis following bovine graft duraplasty for Chiari malformation Type I repair: case report.

Eosinophilic meningitis is a known complication of duraplasty, including that using bovine tissues. Previous authors have relied on surgical removal o...
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