Journal Pre-proof Lumbar subdural hematoma detected after surgical treatment of chronic intracranial subdural hematoma: a case report. Tomoaki Fujita, M. D., Yoshihiro Iwamoto, M. D., Ph. D., Hayato Takeuchi, M. D., Ph. D., Hitoshi Tsujino, M. D., Naoya Hashimoto, M. D., Ph. D. PII: S1878-8750(19)32891-8 DOI: https://doi.org/10.1016/j.wneu.2019.11.053 Reference: WNEU 13722 To appear in: World Neurosurgery Received Date: 10 August 2019 Revised Date: 7 November 2019 Accepted Date: 8 November 2019 Please cite this article as: Fujita T, Iwamoto Y, Takeuchi H, Tsujino H, Hashimoto N, Lumbar subdural hematoma detected after surgical treatment of chronic intracranial subdural hematoma: a case report., World Neurosurgery (2019), doi: https://doi.org/10.1016/j.wneu.2019.11.053. This is a PDF file of an article that has undergone enhancements after acceptance, such as the addition of a cover page and metadata, and formatting for readability, but it is not yet the definitive version of record. This version will undergo additional copyediting, typesetting and review before it is published in its final form, but we are providing this version to give early visibility of the article. Please note that, during the production process, errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain. © 2019 Elsevier Inc. All rights reserved. Title Lumbar subdural hematoma detected after surgical treatment of chronic intracranial subdural hematoma: a case report. Author names and affiliations. Tomoaki Fujita (M. D.) Department of Neurosurgery, Kyoto Yamashiro General Medical Center, Kizuekimae 1-27, Kizugawa-shi, Kyoto 619-0214 Japan friendautumn@gmail.com Yoshihiro Iwamoto (M. D., Ph. D.) Department of Neurosurgery, Kyoto Yamashiro General Medical Center, Kizuekimae 1-27, Kizugawa-shi, Kyoto 619-0214 Japan dr1401@yamashiro-hp.jp Hayato Takeuchi (M. D., Ph. D.) Department of Neurosurgery, Kyoto Yamashiro General Medical Center, Kizuekimae 1-27, Kizugawa-shi, Kyoto 619-0214 Japan thayato@koto.kpu-m.ac.jp Hitoshi Tsujino (M. D.) Department of Neurosurgery, Kyoto Yamashiro General Medical Center, Kizuekimae 1-27, Kizugawa-shi, Kyoto 619-0214 Japan htj-1104@hotmail.co.jp Naoya Hashimoto (M. D., Ph. D.) Department of Neurosurgery, Kyoto Prefectural University Graduate School of Medicine, Kawaramachi-Hirokoji, Kamigyo-ku, Kyoto 602-8566, Japan nhashimo@koto.kpu-m.ac.jp Key words: intracranial chronic subdural hematoma, magnetic resonance imaging, spinal subdural hematoma, bridging vein, migration Running title: Lumbar SSDH after surgical treatment of intracranial CSDH Corresponding author: Tomoaki Fujita, M. D., Department of Neurosurgery, Kyoto Okamoto Memorial Hospital, Sayamanishinoguchi 100, Kumiyama-cho, Kuse-gun, Kyoto 613-0034 Japan Phone: +81-774-48-5500 FAX: +81-774-44-7159 E-mail: friendautumn@gmail.com 1 Abstract 2 Background: 3 Spinal subdural hematoma (SSDH), which can cause lower back pain, leg pain and 4 weakness, is rare, and is usually associated with bleeding tendency, trauma, spinal 5 vascular malformation, intraspinal tumor, or iatrogenic invasion. Only a few cases of 6 SSDH after intracranial chronic subdural hematoma (CSDH) have been reported. We 7 report a case of lumbar SSDH in the absence of predisposing factors after reoperation 8 for recurrent intracranial CSDH that improved with conservative treatment. 9 10 Case Description: 11 Approximately one month after falling, a 63-year-old woman was experiencing left 12 hemiparesis and impaired orientation that was diagnosed as right intracranial CSDH by 13 computed tomography (CT). Surgical treatment for the CSDH led to immediate 14 improvement of her symptoms. On postoperative day 29, the right CSDH recurred with 15 left hemiparesis, and successful reoperation relieved the symptoms within a few hours. 16 One day after the second operation, very small acute subdural hematomas in regions 17 along the left tentorium cerebelli and the left falx cerebri were found on CT. On day 31, 18 she complained of sitting-induced bilateral radiating lower limb pain. Magnetic 19 resonance imaging on day 34 showed an acute SSDH at L4-5 level and a sacral 20 perineural cyst filled with hematoma, although her radiating pain tended to improve. 21 She was treated conservatively and discharged without symptoms on day 44. 22 23 Conclusions: 24 Although SSDH is rare, it is important for neurosurgeons and physicians to consider the 25 possibility of SSDH when lower limb pain or paresis occur after procedures with rapid 26 intracranial pressure alteration such as drainage for intracranial CSDH. 27 28 I. Introduction Intracranial chronic subdural hematoma (CSDH) is a relatively common disease, 29 with an incidence of 8.2/100,000/year after 70 years of age1). In comparison, spinal 30 subdural hematoma (SSDH) is rare, and is usually associated with bleeding diatheses 31 due to hematologic disease or anticoagulation therapy, trauma, iatrogenic injury such as 32 lumbar puncture or previous spinal surgery, spinal vascular malformation, or intraspinal 33 tumor3, 22, 23). Equally as rare is, SSDH associated with intracranial CSDH. SSDH with 34 CSDH can be divided into 2 types; concomitant SSDH and CSDH, and SSDH 35 developing subsequent to CSDH. To our knowledge, only two cases of SSDH after 36 surgery for intracranial CSDH have been reported6, 11). Pathogenesis of it is unknown, 37 but a hypothesis that gravitational migration of intracranial subdural hematoma to form 38 hematoma in the spinal canal is conceivable9, 13, 14, 18). We surmise that the intracranial 39 subdural hematoma of this case was not residual CSDH but newly formed one caused 40 by tearing of infratentorial bridging vein. Here, we report a case of SSDH that induced 41 lower limb pain after surgery for intracranial CSDH but spontaneously resolved with 42 careful conservative observation. 43 II. Case Description 44 A 63-year-old woman with a history of asthma had undergone coil embolization 45 of an left unruptured internal carotid artery aneurysm about ten years previously. She 46 fell while walking and suffered a left wrist fracture, which was repaired orthopedically 47 at another hospital. She did not hit her head, hip or lumbar area. The fracture healed 48 well, but she noticed gradual worsening of gait disturbance. She presented to our 49 outpatient department approximately one month after the fall with impaired orientation 50 and left hemiparesis. Neurological examination revealed a Glasgow Coma Scale of 14 51 (eye opening 4, verbal response 4, motor response 6) and grade 4 in Manual Muscle 52 Testing in her left side. She was not on anticoagulation therapy, and there was no 53 laboratory evidence of a bleeding diathesis. A cranial computed tomography (CT) scan 54 on admission (day 0) showed right CSDH that was causing a right-to-left midline shift 55 of the brain. Burr hole surgery for irrigation and drainage of the CSDH was performed 56 the same day, and her symptoms improved immediately after the operation. She was 57 discharged on day 7. However, left hemiparesis recurred on day 29, and recurrence of 58 intracranial CSDH was confirmed by CT. Re-irrigation and drainage of CSDH was 59 successfully performed the same day, and the symptoms improved within hours. A 60 routine CT check on the day after reoperation (day 30) revealed very small acute 61 subdural hematomas, less than 3 mm in thickness, along the tentorium cerebelli and the 62 falx cerebri on the contralateral (left) side (Fig. 1c, d), which were not identified on CT 63 images just after the reoperation (Fig. 1a, b). Because she did not report headache, and 64 no other subjective symptoms or neurological deficits were present, in addition to the 65 small hematoma volume, we decided to treat her with watchful observation, and the 66 drainage tube was removed from the subdural space the same day. On day 31, she 67 complained for the first time of radiating pain in the posterior surface of both lower 68 limbs when sitting (Straight Leg Raising Test: right 45 degrees and left 45 degrees). 69 Lumbar Magnetic resonance imaging (MRI) on day 34 showed an acute SSDH at the 70 L4-5 level and a perineural cyst filled with hematoma at the sacral level (Fig. 2a, b, 3a, 71 b). On that day, her sitting-induced pain was improving, so she was treated 72 conservatively. On day 37, a cranial CT scan at the level of the tentorium cerebelli and 73 cranial CT scan of the parietal region revealed that a hematoma had migrated (Fig. 1e, 74 f). On day 44, MRI re-examination confirmed spontaneous shrinkage of the hematoma 75 (Fig. 3c, d), so she was again discharged. Follow-up imaging study of the 76 coil-embolized unruptured aneurysm was done before and after treatment period for 77 CSDH. MR angiograms revealed no interval change so the possibility of bleeding from 78 aneurysm could be ignored. We hypothesized that the intracranial subdural hematoma 79 had moved downward to the lumbar spine under gravity, explaining the accumulation of 80 SSDH, and then the hematoma had diffused with cerebrospinal fluid flow, resolving 81 spontaneously. She received outpatient follow-up, and was free of symptoms with no 82 recurrent SSDH (Fig. 3e, f). 83 III. Discussion 84 Spinal hematomas normally present with signs of spinal cord or cauda equina 85 compression, often with an acute onset preceded by back pain or radicular pain4). In 86 cases of spinal hematoma, differentiation between epidural and subdural hematoma is 87 complicated―for example, the image of spinal epidural hematoma reportedly depicts a 88 convex lens-like shape5), or SSDH crowds the cauda equina roots26), but distinguishing 89 them clearly from each other prior to surgery is not easy20). Definitive diagnosis is 90 usually made by operative findings. In this report, we made a diagnosis of SSDH based 91 not on surgical findings but rather on sagittal MRI showing a hematoma distributed 92 along a nerve root. In addition, the signal intensity of SSDH (T1WI-T2WI) was 93 hyper-hyper on day 34 (the 3rd day of lower limb radiating pain), iso-hypo on day 44 94 (the 13th day of lower limb radiating pain), and hypo-hypo on day 77 (the 46th day of 95 lower limb radiating pain), indicating that the hematoma on day 34 was in acute stage4) 96 (Table 1). This finding supports hematoma formation occurring after the second 97 operation, and that the hematoma was not induced by direct injury to the lumbar area 98 when the patient fell. From a clinical point of view, it is consistent that the onset of 99 SSDH was after reoperation because the patient did not complain of radiating pain 100 before the reoperation and her recurrent paresis improved soon after the reoperation. 101 SSDH with CSDH can be divided into 2 types; concomitant SSDH and CSDH, 102 and SSDH developing after CSDH. One prospective study reported a 1.2% occurrence 103 of concomitant SSDH and CSDH 8), and indicated that SSDH may be associated with 104 direct lumbar trauma. However, only two cases of SSDH after CSDH have been 105 reported (Table 2) 6, 11). SSDH is usually attributed to bleeding diatheses due to 106 hematologic disease or anticoagulation therapy, trauma, iatrogenic injury such as 107 lumbar puncture or previous spine surgery, spinal vascular malformation, or intraspinal 108 tumors3, 22, 23), but in this case no such pathogenesis was detected. It is very rare for 109 iatrogenic SSDH to be detected after intracranial surgery2, 6, 10, 11, 16, 17, 18). 110 Assessing the pathogenesis of SSDH after intracranial surgery, some authors have 111 hypothesized that transmission of surgery-associated cerebrospinal fluid (CSF) 112 hypotension into the spinal subdural compartment may result in rupture of the 113 intraspinal vasculature2, 10, 16, 17). Another theory assume opening of dura-arachnoid 114 space due to shrunken brain damage small anastomosing network of spinal dural 115 vessels, and that cause SSDH.7) In general, however, the subdural space of the spine is 116 poorly vascularized. Additionally, electron microscopy21) and 117 pneumoencephalography19) reveal anatomical continuity between subdural spaces 118 within the skull and the spinal canal. Therefore, to date, the most plausible hypothesis is 119 that an intracranial subdural hematoma migrates downward under gravity to form a 120 hematoma in the spinal canal9,13, 14, 18). Where the hematoma moved from in our case is 121 an important issue. Because intracranial CSDH is enveloped by the inner and outer 122 membranes, it is hard to comprehend how the contents of CSDH could move from the 123 intracranial space to another location25). In our case, cranial CT detected very small 124 acute subdural hematomas in contralateral regions along the tentorium cerebelli and 125 the falx cerebri on day 30, one day after the reoperation, although they were not 126 detected on CT just after the reoperation. In recent growing consensus, drainage after 127 preoperative intracranial hypertension would result in transtentorial pressure gradient to 128 tearing of bridging veins in posterior fossa or remote cerebellar hemorrhage 15). 129 Likewise, we guessed that a bridging vein was torn due to brain-shifting caused by 130 overnight drainage of CSDH12, 24). Thus, instead of residual CSDH, we speculate that 131 freshly-formed subdural hematomas became distributed to the subdural space of the 132 lumbar spinal canal and even partially into a sacral perineural cyst. As a result, an acute 133 lumbar subdural hematoma manifested, causing sitting-induced lower limb pain two 134 days after reoperation. It is unclear how SSDH can develop in very limited cases of 135 CSDH but not in the majority, and further accumulation of cases will be necessary to 136 understand the mechanism. In the present patient, the hematoma resolved and symptoms 137 disappeared with conservative treatment. Spontaneous improvement was also seen after 138 conservative treatment in most of the cases in previous reports. However, prompt spinal 139 tap or even surgery to evacuate the hematoma may be indicated if there is progressive 140 neurologic decline18). Thus, careful and close neurological observation is mandatory 141 when a patient complain spinal symptoms after cranial surgery. 142 143 IV. Conclusions We reported a case of symptomatic lumbar SSDH induced possibly by migration 144 of an intracranial subdural hematoma after surgical treatment for CSDH. The SSDH 145 resolved with conservative treatment. However, in some cases, there can be progressive 146 neurologic decline and surgery may be required. SSDH is a rare complication after 147 cranial surgery, but neurosurgeons should consider the possibility of SSDH when lower 148 limb pain and/or paresis manifest after surgery involving a rapid alteration of 149 intracranial pressure. 150 References 151 1. 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J Formos Med Assoc. 2009; 108: 224 258-61. 225 226 Figure legends 227 Fig. 1 228 (a) Cranial CT scan at the level of the tentorium cerebelli on postoperative day 0 and (b) 229 cranial CT scan of the parietal region on postoperative day 0 show the replacement of 230 CSH with saline solution and air. 231 (c) Cranial CT scan at the level of the tentorium cerebelli on postoperative day 1 and (d) 232 cranial CT scan of the parietal region on postoperative day 1 show very small acute 233 subdural hematomas undetected on postoperative day 0 in regions along the tentorium 234 cerebelli (→) and the falx cerebri (⇢) on the left side. 235 (e) Cranial CT scan at the level of the tentorium cerebelli on postoperative day 7 and (f) 236 cranial CT scan of the parietal region on postoperative day 7 show a hematoma had 237 migrated elsewhere. 238 239 Fig. 2 240 (a) T1W axial image and (b) T2W axial image at the lumbosacral level on postoperative 241 day 5 show a bilateral subdural hematoma (→). 242 243 Fig. 3 244 (a) T1W sagittal image and (b) T2W sagittal image at the lumbosacral level on 245 postoperative day 5 (the 3rd day of lower limb pain) show iso-hypo signal intensity of 246 hematoma on T1WI-T2WI. The hematoma (→) is distributed along a nerve root, and a 247 perineural cyst (⇢) is filled with hematoma at the sacral level. 248 (c) T1W sagittal image and (d) T2W sagittal image at the lumbosacral level on 249 postoperative day 15 (the 13th day of lower limb pain) show hyper-hyper signal intensity 250 of hematoma on T1WI-T2WI. 251 (e) T1W sagittal image and (f) T2W sagittal image at the lumbosacral level on 252 postoperative day 48 (the 46th day of lower limb pain) show hypo-hypo signal intensity 253 of hematoma on T1WI-T2WI. 254 The signal intensity changes indicate that an acute subdural hematoma in the lumbar 255 spine is becoming chronic, while the hematoma in a perineural cyst is reducing in size. 256 257 Table 1 258 An excerpt from reference 2. a) Timing of the 1st lumbar spine MRI. b) Timing of the 259 2nd lumbar spine MRI. c) Timing of the 3rd lumbar spine MRI. 260 261 Table 2 262 Summary of three previous reported cases of SSDHs after surgery for CSDH in the 263 literature. 264 Table 1 An excerpt from reference 2. a) Timing of the 1st lumbar spine MRI. b) Timing of the 2nd lumbar spine MRI. c) Timing of the 3rd lumbar spine MRI. Table 2 Summary of three previous reported cases of SSDHs after surgery for CSDH in the literature. Phase Hyperacute Acute Age Hemoglobin T1-T2 12 Oxyhemoglobin Iso-hyper 1-3 days Deoxyhemoglobin Hypo-hypo ←a Early subacute 3-7 days Intracellular methemoglobin Hyper-hypo Late subacute 1-2 weeks Extracellular methemoglobin Hyper-hyper ←b Hemosiderin Hypo-hypo ←c Chronic 2 weeks MR appearance of intraspinal hematoma Table 1 An excerpt from reference 4. a) Timing of the 1st lumbar spine MRI. b) Timing of the 2nd lumbar spine MRI. c) Timing of the 3rd lumbar spine MRI. Authors Age years, SSDH level Symptom Onset Symptoms Treatment Outcome 3 days Low back pain, leg Surgical evacuation Complete recovery Sex Lee et al., 68, female L4-S1 11) 2007 Hagihara et paresthesia 47, man L3-S1 2 months Low back pain Conservative Complete recovery 63, female L4-5 2 days Lower limb Conservative Complete recovery 6) al.,2010 Present case radiating pain Cases of SSDHs after surgery for CSDH Table 2 Summary of three previous reported cases of SSDHs after surgery for CSDH in the literature.