REGIONAL ANESTHESIA AND ACUTE PAIN

CASE REPORT

Acute Vision Loss Secondary to Epidural Blood Patch Terson Syndrome Gabriel L. Pagani-Estévez, MD,* John J. Chen, MD, PhD,† James C. Watson, MD,*‡ and Jacqueline A. Leavitt, MD† Background and Objectives: Lumbar epidural blood patch (EBP) is a commonly used procedure to treat postdural puncture headache. We present a case of vision loss immediately following an EBP. Case Report: A 49-year-old woman with idiopathic intracranial hypertension received an EBP for postdural puncture headache at an outside facility without fluoroscopic guidance and in the seated position. The patient experienced syncope during the procedure as 25 mL of autologous blood was rapidly injected. The patient regained consciousness and described bilateral vision loss. Brain magnetic resonance imaging was negative for hemorrhage. Dilated fundus examination revealed significant, bilateral retinal and vitreous hemorrhage consistent with Terson syndrome. Conclusions: This patient developed Terson syndrome as an immediate EBP complication. Iatrogenic Terson syndrome has been previously described with epidural space saline and anesthetic injections, but not EBP. Of 11 reported cases, 10 were female, and 9 had complete vision recovery. Previous studies have demonstrated that epidural space injection increases subarachnoid pressure in a volume- and rate-dependent fashion. An abrupt increase in subarachnoid space pressure likely led to retinal hemorrhage by compromising retinal venous drainage. This is the first known case of Terson syndrome caused by EBP. Injectate volume should be minimized, and a slow rate of injection pursued. The anesthesiologist, pain interventionist, and ophthalmologist should be aware of this rare but disabling complication and consider taking extra precautions when consenting patients for EBP with vision compromise or comorbidities concerning for elevated intracranial pressure. (Reg Anesth Pain Med 2016;41: 164–168)

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umbar epidural blood patch (EBP) is a commonly used procedure to treat postdural puncture headache. First performed in 1960, it is estimated to have a success rate between 60% and 95% with a low rate of complications.1 Local complications include pain, paresthesia, infection, and hematoma. Even more rarely, bradycardia, loss of consciousness, cranial nerve palsies, seizures, and cerebral ischemia presumed related to vasospasm have been described. Although the loss-of-resistance technique is universally used, the additional use of fluoroscopy with contrast administration can be used to confirm needle tip placement in the epidural space. We present a case of vision loss immediately following an EBP.

From the Departments of *Neurology, †Neuro-Ophthalmology, and ‡Anesthesiology, Mayo Clinic, Rochester, MN. Accepted for publication October 13, 2015. Address correspondence to: Gabriel L. Pagani-Estévez, MD, Department of Neurology, Mayo Clinic, 200 1st St SW, Rochester, MN 55905 (e‐mail: [email protected]). This work was supported in part by an unrestricted grant to the Mayo Clinic Department of Ophthalmology by Research to Prevent Blindness, Inc, New York, NY. The authors declare no conflict of interest. Copyright © 2016 by American Society of Regional Anesthesia and Pain Medicine ISSN: 1098-7339 DOI: 10.1097/AAP.0000000000000352

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CASE REPORT Patient permission was verbally obtained from the patient in principle for the publication of this study. Written permission was obtained for the use of the patient's medical records. A 49-year-old woman from an outside institution presented with vision loss immediately following an EBP for a postdural puncture headache. She had a remote history of idiopathic intracranial hypertension, which had been clinically inactive for many years. There was also a known left sphenoid wing meningioma treated 14 years previously with gamma knife radiosurgery. In this context, she had presented for evaluation of recurrent, chronic daily headache. Given her history of idiopathic intracranial hypertension, a lumbar puncture was scheduled. Three days before her blood patch, she had a prone lumbar puncture collecting 3 mL of clear, colorless fluid. The cerebrospinal fluid parameters were normal (opening pressure 145 mm, 0 white blood cells/μL, 798 red blood cells/μL, and 28 mg/dL of protein), and there were no reported procedural difficulties. However, within hours, the patient developed a different headache phenotype consistent with an orthostatic postdural puncture headache. An EBP was arranged. The EBP was performed with the patient seated and without fluoroscopic guidance. The patient had an 18-gauge Tuohy needle placed into the L3/L4 epidural space. Negative aspiration of cerebrospinal fluid and blood and absence of lower-extremity paresthesia were documented. Ten milliliters of autologous blood was injected into the epidural space immediately after which the patient experienced syncope. The speed of injection was not documented. The syncope was a presumed vasovagal reaction, and normal vital signs were documented. As steps were taken to resuscitate the patient, an additional 15 mL of autologous blood was rapidly injected. It was unclear whether the needle position in the epidural space had been reconfirmed. The patient regained consciousness quickly but immediately noted bilateral vision loss described as “red holes obstructing [her] vision.” She presented to an emergency department the following day, and a gadolinium-enhanced brain magnetic resonance imaging demonstrated known changes consistent with her prior diagnoses: a stable-appearing left cavernous sinus meningioma and stable imaging stigmata consistent with her history of idiopathic intracranial hypertension (mild prominence of subarachnoid fluid surrounding the bilateral optic nerves and a partially empty sella turcica). There was no evidence of hemorrhage in the orbit, subarachnoid space, or brain parenchyma. There were no foci of restricted diffusion concerning for ischemia and no abnormal meningeal enhancement. Five days later, ophthalmologic evaluation revealed the ability to count fingers at only 5 f. in the right eye (OD) and a best corrected Snellen visual acuity of 20/100 in the left eye (OS). Her baseline visual acuity had been 20/20 OD and 20/70 OS. Intraocular pressure was 17 mm Hg bilaterally on tonometry. Extraocular movements were full, and there was no relative afferent pupillary defect. Dilated fundus examination revealed significant, multiple, round blot hemorrhages in the macula involving both the subretinal and intraretinal spaces (Fig. 1). In addition,

Regional Anesthesia and Pain Medicine • Volume 41, Number 2, March-April 2016

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EBP and Terson Syndrome

FIGURE 1. Fundus photographs demonstrate significant left eye (top) and right eye (bottom) preretinal and subretinal hemorrhage involving primarily the macula. The optic discs have mild peripapillary atrophy with distinct margins and absence of pallor. Tortuous retinal vessels are present in both eyes.

there was mild vitreous hemorrhage present. The retinal vessels were mildly tortuous. The optic discs had no papilledema or pallor. The patient presented to our institution 14 days later for a second opinion, given her lack of significant visual improvement. At that time, Snellen visual acuity had improved to 20/400 OD and 20/70 OS. Dilated eye examination showed persistent © 2016 American Society of Regional Anesthesia and Pain Medicine

preretinal and subretinal hemorrhages OD and subretinal hemorrhages OS. Spectralis macular optical coherence tomography confirmed hemorrhages in the fovea and parafoveal regions, involving the superficial and deep retina (Fig. 2). The component of vitreous hemorrhage was not felt severe enough to justify vitrectomy as an attempt to hasten visual recovery.

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DISCUSSION This patient developed Terson syndrome as an immediate complication of her lumbar EBP procedure. Terson syndrome was originally described in 1900 as an oculocerebral syndrome of retinal and vitreous hemorrhage associated most commonly with aneurysmal subarachnoid hemorrhage. In fact, its clinical presence is a poor prognostic factor among aneurysmal subarachnoid hemorrhage patients.2 The condition has also been described in trauma, subdural hematoma, arteriovenous malformation, acute promyelocytic leukemia, and strangulation.3 Iatrogenic Terson syndrome has also been described with epidural space injection.4–10 Eleven cases total have been reported in the literature following lumbar spine epidural saline and steroid injection (Table 1). Ten of those 11 were female, and full visual recovery took place in 9 of 11 patients over a time span ranging from 3 months to 3 years. The volume of injectate was 20 mL in 1 case but was typically higher, varying from 40 to 120 mL in the remaining cases. The injectate was either saline or a combination of saline and local anesthetic with steroid admixed. In a few cases, the injectate was documented as being administered slowly. Four cases were performed under general anesthesia, and the use of certain anesthetics, particularly inhalational anesthetics such as halothane, may elevate intracranial pressure (ICP) through cerebral vasodilation and increased cerebral blood flow.11 Fluoroscopic imaging guidance was not used in these cases, and confirmation of needle position in the epidural as opposed to subdural space was not routinely documented. None of the reported cases of lumbar epidural injection involved a loss of consciousness or use of autologous blood. The pathophysiology in this case and prior cases of saline epidural injections likely involved a rapid increase in subarachnoid space pressure. A study using 24 human subjects in the

lateral, flexed position measured epidural and subarachnoid compartment pressures following the injection of 10 to 20 mL of fluid into the epidural space.12 Sacral canal injection did not increase subarachnoid space pressures and barely increased epidural space pressure. Lumbar injections, however, caused an abrupt increase in both compartments with epidural pressures reaching a peak of 65 cm H2O and subarachnoid space pressures reaching a maximum of 85 cm H2O. Increased volume and a more rapid rate of infusion correlated with increased pressure readings. Injection of 15 mL of blood into the epidural space was also separately shown to cause an immediate increase of more than 3-fold in subarachnoid pressure.13 The optic nerve is surrounded by an extension of the subarachnoid space, and the central vein of the retina crosses this space to drain into the ophthalmic veins and cavernous sinus. Venous drainage from the eye therefore depends on venous pressure of the central retinal vein exceeding the subarachnoid space pressure. An abrupt increase in the subarachnoid space pressure can lead to retinal venous and capillary hemorrhage and through choroidal anastomotic channels can result in vitreous hemorrhage. Because autologous blood was injected in our patient, an alternative mechanism could have involved inadvertent instillation of injectate into the subarachnoid space with subsequent tracking of blood along the optic nerve sheath, lamina cribrosa, and into the bilateral retinae.14 This latter mechanism, of course, cannot account for Terson syndrome caused by epidural saline injection, and the vitreous humor does not communicate with the subarachnoid space. This is the first known reported case of Terson syndrome following an EBP injection. It is possible that the patient's history of idiopathic intracranial hypertension predisposed her to this condition. It is also possible that her syncope resulted from an abrupt

FIGURE 2. Spectralis macular optical coherence tomography showing deep retinal hemorrhages (solid lines) and superficial hemorrhages (dashed lines) in both the right (top) and left (bottom) eyes.

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© 2016 American Society of Regional Anesthesia and Pain Medicine

F F F F F

F

F

M F

F

F

35 35 36† 39† 39

43†

47†

47 49

53

81

200 mg HC in 0.25% bupivacaine 80 mg DM in 0.25% bupivacaine “Steroid” in 0.9% NaCl 80 mg MP in 0.125% bupivacaine 80 mg MP in 0.5% lidocaine 80 mg MP in 0.5% lidocaine

80 mg MP in 0.5% lidocaine 80 mg MP in 0.5% lidocaine 0.9% NaCl 0.9% NaCl 0.9% NaCl

Medication Injected

41 mL

41 mL

ND 20 mL

40 mL

40 mL

41 mL 41 mL 120 mL 120 mL 100 mL

Injectate Volume

1 mL/s

1 mL/s

ND 1 min

ND

2 min

1 mL/s 1 mL/s ND ND “Brisk”

Injection Rate

OU RH

OS multiple, macular RH

OS VH OU macular RH

OU macular RH and PH

OU RH, PH, VH

OS multiple RH OD PH OD multiple PH, RH OD macular RH; OS, VH OD PH, VH; OS RH

Funduscopic Examination

Visual Deficit

20/200 OD; 20/50 OS

20/30 OD; 20/25 OS

20/20 OD; 20/200 OS 20/400 OD; 20/40 OS

20/200 OU

20/30 OU 20/25 OD; 20/20 OS OD blurring Light perception alone “Hand motions” OD; 20/200 OS OD blindness

Kushner and Olson,7 1995

Improved, with “blind spots” at 3 mo 20/50 OD; 20/40 OS at 5 mo‡

Kushner and Olson,7 1995

Gibran et al,10 2002 Purdy and Ajimal,8 1998 20/20 at 36 mo§ 20/40 OU at 24 mo

Ling et al,6 1993

Victory et al,5 1991

OD “normal” at 1.5 mo OD 20/20, OS 20/60 at 3 mo‡

Reference Kushner and Olson, 19957 Kushner and Olson,7 1995 Clark and Whitwell,4 1961 Clark and Whitwell,4 1961 Naseri et al,9 2001

Vision Outcome* Resolution in “a few” months Complete recovery in 1.5 mo OD 20/80 OS 20/17 at 36 mo OD 20/17 OS 20/20 at 5 mo 20/20 in “a few” months

*Visual acuity reported in the British fashion was converted to the American system for ease of interpretation. †These 4 patients received general anesthesia during their epidural injection. The 36- and 39-year-old patients did not have the general anesthesia regimen described. The 43-year-old patient received thiopentone, nitrous oxide in oxygen, and isoflurane, and the 47-year-old patient received propofol and nitrous oxide in oxygen. ‡These were specifically documented as the premorbid baseline visual acuity, indicating complete visual recovery. §This patient underwent an OS vitrectomy after 36 months with complete vision recovery subsequently. DM indicates depomedrone; F, female; HC, hydrocortisone; M, male; MP, methylprednisolone; ND, not documented; OD, right eye; OS, left eye; OU, both eyes; PH, preretinal hemorrhage; RH, retinal hemorrhage; VH, vitreous hemorrhage.

Sex

Age, y

TABLE 1. Literature Review of Case Reports Describing Terson Syndrome From Epidural Injection

Regional Anesthesia and Pain Medicine • Volume 41, Number 2, March-April 2016 EBP and Terson Syndrome

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increase in ICP and not a vasovagal reaction.3 The seated position may increase ICP through Valsalva and increased intra-abdominal pressure and should potentially be avoided during an EBP.15 This case illustrates that the minimum volume possible of injectate should be used, and a very slow rate of injection pursued to avoid iatrogenic Terson syndrome. The anesthesiologist, pain interventionist, and ophthalmologist should be aware of this rare, but disabling, complication and consider taking extra precautions when consenting patients with preexisting poor vision, 1 eye, or comorbidities concerning for elevated ICP. REFERENCES 1. Kalina P, Craigo P, Weingarten T. Intrathecal injection of epidural blood patch: a case report and review of the literature. Emerg Radiol. 2004;11: 56–59. 2. Sung W, Arnaldo B, Sergio C, Juliana S, Michel F. Terson's syndrome as a prognostic factor for mortality of spontaneous subarachnoid haemorrhage. Acta Ophthalmol. 2011;89:544–547. 3. Hassan A, Lanzino G, Wijdicks EF, Rabinstein AA, Flemming KD. Terson's syndrome. Neurocrit Care. 2011;15:554–558.

6. Ling C, Atkinson PL, Munton CG. Bilateral retinal haemorrhages following epidural injection. Br J Ophthalmol. 1993;77:316–317. 7. Kushner FH, Olson JC. Retinal hemorrhage as a consequence of epidural steroid injection. Arch Ophthalmol. 1995;113:309–313. 8. Purdy EP, Ajimal GS. Vision loss after lumbar epidural steroid injection. Anesth Analg. 1998;86:119–122. 9. Naseri A, Blumenkranz MS, Horton JC. Terson's syndrome following epidural saline injection. Neurology. 2001;57:364. 10. Gibran S, Mirza K, Kinsella F. Unilateral vitreous haemorrhage secondary to caudal epidural injection: a variant of Terson's syndrome. Br J Ophthalmol. 2002;86:353–354. 11. Bazin JE. Effects of anesthetic agents on intracranial pressure [in French]. Ann Fr Anesth Reanim. 1997;16:445–452. 12. Usubiaga JE, Usubiaga LE, Brea LM, Goyena R. Effect of saline injections on epidural and subarachnoid space pressures and relation to postspinal anesthesia headache. Anesth Analg. 1967;46:293–296. 13. Coombs DW, Hooper D. Subarachnoid pressure with epidural blood “patch”. Reg Anesth Pain Med. 1979;4:3–6.

4. Clark CJ, Whitwell J. Intraocular haemorrhage after epidural injection. Br Med J. 1961;2:1612–1613.

14. Ogawa T, Kitaoka T, Dake Y, Amemiya T. Terson syndrome: a case report suggesting the mechanism of vitreous hemorrhage. Ophthalmology. 2001;108:1654–1656.

5. Victory RA, Hassett P, Morrison G. Transient blindness following epidural analgesia. Anaesthesia. 1991;46:940–941.

15. Lee SC, Lueck CJ. Cerebrospinal fluid pressure in adults. J Neuroophthalmol. 2014;34:278–283.

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Acute Vision Loss Secondary to Epidural Blood Patch: Terson Syndrome.

Lumbar epidural blood patch (EBP) is a commonly used procedure to treat postdural puncture headache. We present a case of vision loss immediately foll...
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