Pathogenesis of spinal chronic subdural hematoma based on histopathological findings: a case report

Pathogenesis of spinal chronic subdural hematoma based on histopathological findings: a case report | Research Square window.SnipcartSettings = { analytics: { enabled: false } }; (function() { var accessVector = localStorage.getItem('access_vector') || ''; window.dataLayer = window.dataLayer || []; if (accessVector) { window.dataLayer.push({ user: { profile: { profileInfo: { snid: accessVector } } } }); } })(); (function(w,d,s,l,i){w[l]=w[l]||[];w[l].push({'gtm.start':new Date().getTime(),event:'gtm.js'});var f=d.getElementsByTagName(s)[0],j=d.createElement(s),dl=l!='dataLayer'?'&l='+l:'';j.async=true;j.src='https://www.googletagmanager.com/gtm.js?id='+i+dl;f.parentNode.insertBefore(j,f);})(window,document,'script','dataLayer','GTM-K279D39R'); Browse Preprints In Review Journals COVID-19 Preprints AJE Video Bytes Research Tools Research Promotion AJE Professional Editing AJE Rubriq About Preprint Platform In Review Editorial Policies Our Team Advisory Board Help Center Sign In Submit a Preprint Cite Share Download PDF Case Report Pathogenesis of spinal chronic subdural hematoma based on histopathological findings: a case report Yoshiki Fujikawa, Takahiro Fujishiro, Hideki Tanabe, Toshihiro Takami, and 1 more This is a preprint; it has not been peer reviewed by a journal. https://doi.org/ 10.21203/rs.3.rs-4295510/v1 This work is licensed under a CC BY 4.0 License Status: Posted Version 1 posted You are reading this latest preprint version Abstract Background Spinal chronic subdural hematoma (sCSDH) is a rare condition whereas intracranial chronic subdural hematoma (iCSDH) is well-recognised and documented in clinical settings. Despite various theories have been proposed, the exact pathogenesis of sCSDH remains to be elucidated. Herein, we report a rare case of sCSDH with co-existing iCSDH and deduce its etiology using histopathological findings. Case presentation A 76-year-old Japanese man with slight hemiparesis due to iCSDH underwent burr hole surgery with successful drainage of the hematoma. The patient could not walk unassisted after the surgery. Spinal magnetic resonance imaging (MRI) was performed, revealing a subdural hematoma extending from the T11 to S2 levels. The patient decided to undergo hematoma evacuation via a hemilaminectomy. Intraoperatively, we identified a dark liquefied hematoma with its capsule, which closely resembled an iCSDH. Histological examination of the resected capsule revealed loose fibrovascular tissues comprising capillaries, collagen fibres, small number of macrophages, and hemosiderin deposits. These findings were very similar to those for iCSDH, such that the pathogenesis of sCSDH was considered identical to that of iCSDH. The patient was transferred to a rehabilitation hospital, and 4 months after the lumbar surgery, no recurrence was observed in MRI examination. Conclusions sCSDH may result from various potential pathogeneses, that clinicians should consider for correct diagnosis and appropriate management. Our case provides valuable insights into the pathogenesis of sCSDH. Chronic subdural hematoma Histopathology Pathogenesis Spinal subdural hematoma Figures Figure 1 Figure 2 Figure 3 Figure 4 Figure 5 Figure 6 Background Subdural hematoma (SDH) often occurs in the intracranial space, whereas its occurrence along the spinal column in the extracranial space is rare [1, 2]. When presenting symptoms such as lower back pain, weakness in the lower extremities, or bowel and bladder dysfunction that persist for more than 2 weeks, it is classified as chronic SDH (CSDH) [3]. In the spine, the frequency of occurrence of CSDH is considerably lower than that of acute or subacute SDH [4]. Previous studies have proposed various factors contributing to spinal CSDH (sCSDH), including a history of trauma, lumbar puncture, bleeding diathesis, use of antithrombotics, or vascular malformation, although instances of spontaneous occurrence have been noted [5]. In addition, various cases of concomitant intracranial CSDH (iCSDH) and sCSDH have been reported. However, the exact aetiology and pathogenesis of sCSDH remain unknown. In addition, few reports have considered histopathological findings in investigating the pathogenesis of sCSDH. Herein, we present a case of concomitant iCSDH and sCSDH and discuss the pathogenesis of the disease based on the findings of histopathological examination. Case Presentation A 76-year-old man, who had suffered from head trauma 1 month ago, was transferred to our hospital after falling and sustaining head trauma again. He had a history of brain infarction a few decades prior to admission and had undergone burr hole surgery for a right iCSDH 4 years earlier. He was not on anticoagulant or antiplatelet medication. He presented with slight right hemiparesis (manual muscle test; MMT 4/5). Head computed tomography (CT) revealed a left iCSDH with a mild midline shift (Fig. 1A). He underwent burr hole surgery upon admission to our hospital, resulting in successful drainage of the hematoma (Fig. 1B). However, he remained unable to stand or walk unassisted 1 week after surgery. The MMT score for his legs was approximately 3 to 4, and his patellar tendon reflex was attenuated. The Babinski reflex was negative. Spinal magnetic resonance imaging (MRI) revealed a subdural hematoma extending from the T11 to S2 levels (Fig. 2A–D), with no evidence of hematoma at the cervical level (Fig. 2E, F). To improve his neurological condition, he underwent an L2–4 hemilaminectomy for hematoma evacuation. When the dura matter was opened, a dark-brownish liquid hematoma covered by the membrane resembling that of an iCSDH was observed (Fig. 3A, B). Subsequently the hematoma was washed and evacuated through saline irrigation using a drainage tube. Clear cerebrospinal fluid (CSF) was observed through the intact, normal arachnoid membrane, with no evidence of hemorrhage in the subarachnoid space (Fig. 3C). Histopathological examination revealed that the membrane of the hematoma contained fibroblasts, collagen fibres, capillaries, macrophages, and hemosiderin (Fig. 4A, B). Neither hemorrhage nor apparent cell infiltration was observed in the membrane. The patient was transferred to a rehabilitation hospital 9 days after lumbar surgery. Because of dementia, he lacked the willingness to rehabilitate, resulting in him being forced to use a wheelchair to move at discharge. Postoperative spinal MRI at 4 months showed no recurrence of subdural hematoma (Fig. 5A–D). Discussion and Conclusions In this study, we report a rare case of concomitant iCSDH and sCSDH and the histopathological examination of its membrane. To the best of our knowledge, this is the first report to provide histopathological evidence of concomitant sCSDH and iCSDH. Intracranial SDH occurs spontaneously or is triggered predominantly by trauma [6-8], followed by intracranial hypotension [9]. Intracerebral hemorrhage [10, 11], ruptured cerebral aneurysm [12, 13], and cerebral vascular malformations [14-17] are occasionally accompanied by SDH, when bleeding extends to the subdural space. Other less common causes include vasculopathy [18] and neoplasm [19-21]. Development of iCSDH typically occurs as follows: after development of an acute SDH, the absorption of blood subsequently begins with breakdown of erythrocytes and other cellular components. Next, collagen synthesis is induced, and fibroblasts spread over the inner surface of the dura to form a thick outer membrane [22]. Subsequently, a thinner inner membrane forms, leading to the complete encapsulation of the clot. In general, this process spans approximately 2 weeks. The exact pathogenesis of sCSDH remains unknown (Fig. 6), with various theories proposed. One theory suggests that disruption of bridging veins in the spine, caused by direct trauma or lumbar puncture, leads to hematoma formation, similar to intracranial hematomas. The spinal subdural space, known to be less vascularised [23, 24], may contribute to the lower frequency of sCSDH compared to iCSDH. However, bridging veins exist not only in intracranial structures but also in the spine [25], suggesting that disruption of these veins can also cause a spinal SDH. A previous prospective study reported a significant association between direct impact to the lumbar area and spinal SDH in patients with iCSDH, suggesting double trauma to the head and lumbar regions as a major cause of concomitant intracranial and sCSDH. Another theory proposes that an iCSDH might migrate to the spinal area, supported by the less vascularised spinal subdural space and the subdural compartment between the cranium and spinal space proved to be connected [26]. Certain cases have shown hematomas in the posterior fossa or all through the spine [27, 28], with similar signal intensity on MRI, suggesting a common origin. However, our patient did not have a hematoma at the cervical level, as revealed through MRI. Furthermore, the previous prospective study found only 2 (1.2%) of 168 cases of iCSDH with concomitant spinal SDH on MRI [29]. A third theory suggests a different mechanism for the formation of sCSDH from that of iCSDH [30]. In this theory, sCSDH occurs due to a rapid increase in intravascular pressure of the intraspinal segments of lateral spinal vessels, caused by increased abdominal and thoracic pressure. This sudden increase in the intravascular pressure is not neutralised by a simultaneous increase in CSF pressure because of the shielding effect of the spinal column and its ligaments. Consequently, hemorrhage in the subdural space may occur. Such subdural accumulation of blood would be subject to the same set of forces that govern the formation of intracranial fluid collections. This theory is supported by a case report of sCSDH co-existing with a ligament flavum hematoma, indicating a mechanism consistent with this theory [31]. Finally, some researchers have stated that the subarachnoid hemorrhage is thought to extend into the subdural space [32, 33]. These potential pathogeneses may be more likely to be induced by the use of antithrombotics or because of bleeding diathesis. During iCSDH formation, the outer and inner membranes that surround the hematoma form subsequently, as described previously [34]. According to histopathological examination, the outer membrane contains various components, such as neovasculature and fibrous tissue. Exudation from these neovasculatures contributes to hematoma enlargement. Previous studies have categorised histopathological features of the outer membrane into the following four subtypes: noninflammatory (I), inflammatory (II), hemorrhagic inflammatory (III), and scar inflammatory (IV) [35]. The noninflammatory (I) type comprises immature fibroblasts, collagen fibres, sparse cellular infiltration, and neocapillaries. The inflammatory (II) type comprises a single sheet of immature connective tissue with marked cellular infiltration and vascularisation. The hemorrhagic inflammatory (III) type consists of a few sheets of connective tissue with cellular infiltration and moderately-sized capillaries. The scar inflammatory (IV) type comprises inflammatory cell infiltration, neovascularisation, and hemorrhage. In addition, the outer membrane is thought to change from type I to IV in order [36]; thus, the histopathological appearance of the membrane is useful in estimating the duration of CSDH [37]. In our patient, the histopathological features of the outer membrane of the sCSDH were considered to be similar to those of type I iCSDH because of the slight cellular infiltration. Therefore, we suggest that in this case, the mechanism for sCSDH formation may be trauma to the lumbar area, similar to that for iCSDH resulting from head trauma, rather than migration. Moreover, based on the histopathology, this case was in the early phase of hematoma formation. Various studies have also reported that the outer membrane of the sCSDH was observed during surgery [4, 38], similar to our case. On the other hand, some studies did not find an outer membrane [39, 40], which may indicate that not enough time had elapsed for capsular formation or that the hematoma migrated from the cranial space. In conclusion, we demonstrated that sCSDH may have various potential pathogeneses, which can be elucidated through histopathological examination as in our case. This finding provides important insights for clinicians to enable them to correctly diagnose and appropriately manage sCSDH. Abbreviations CSDH, chronic subdural hematoma CSF, cerebrospinal fluid CT, computed tomography iCSDH, intracranial chronic subdural hematoma MMT, manual muscle test MRI, magnetic resonance imaging sCSDH, spinal chronic subdural hematoma SDH, Subdural hematoma Declarations Ethics approval and consent to participate Approval from an ethics review committee was not sought as the publication is a case report. Consent for publication We obtained written informed consent for publication from the patient. Availability of data and materials Not applicable. Competing interests The authors declare that they have no competing interests. Funding None. Authors’ contributions YF, TF, and HT were involved in diagnosis and managing the patient. YF drafted the manuscript while TT critically revised it. MW supervised this study. All authors read and approved the final manuscript. Acknowledgements The authors would like to acknowledge Prof. Seiichi Hirota (Department of Surgical Pathology, Hyogo College of Medicine, Nishinomiya, Hyogo, Japan) for his advice regarding histopathological analysis. We also would like to thank Aya Sunamura and Akemi Usui for their secretarial work and Editage for English language editing. References de Beer MH, Eysink Smeets MM, Koppen H. Spontaneous Spinal Subdural Hematoma. Neurologist. 2017;22 1:34-9; doi: 10.1097/nrl.0000000000000100. Benyaich Z, Laghmari M, Lmejjati M, Aniba K, Ghannane H, Ait Benali S. Acute Lumbar Spinal Subdural Hematoma Inducing Paraplegia After Lumbar Spinal Manipulation: Case Report and Literature Review. World Neurosurg. 2019;128:182-5; doi: 10.1016/j.wneu.2019.05.002. 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[Histological study of the outer membrane of chronic subdural hematoma: possible mechanism for expansion of hematoma cavity]. No Shinkei Geka. 1993;21 8:697-701. Sato S, Suzuki J. Ultrastructural observations of the capsule of chronic subdural hematoma in various clinical stages. J Neurosurg. 1975;43 5:569-78; doi: 10.3171/jns.1975.43.5.0569. Bokka S, Trivedi A. Histopathological study of the outer membrane of the dura mater in chronic sub dural hematoma: Its clinical and radiological correlation. Asian J Neurosurg. 2016;11 1:34-8; doi: 10.4103/1793-5482.154979. Leber KA, Pendl G, Kogler S, Kammerhuber F, Ebner F. Simultaneous spinal and intracranial chronic subdural hematoma. Case illustration. J Neurosurg. 1997;87 4:644; doi: 10.3171/jns.1997.87.4.0644. Kanamaru H, Kanamaru K, Araki T, Hamada K. Simultaneous Spinal and Intracranial Chronic Subdural Hematoma Cured by Craniotomy and Laminectomy: A Video Case Report. In: Case Rep Neurol. Switzerland; 2016. p. 72-7. Matsumoto H, Matsumoto S, Yoshida Y. Concomitant Intracranial Chronic Subdural Hematoma and Spinal Subdural Hematoma: A Case Report and Literature Review. World Neurosurg. 2016;90:706.e1-.e9; doi: 10.1016/j.wneu.2016.03.020. Additional Declarations No competing interests reported. Cite Share Download PDF Status: Posted Version 1 posted You are reading this latest preprint version Research Square lets you share your work early, gain feedback from the community, and start making changes to your manuscript prior to peer review in a journal. As a division of Research Square Company, we’re committed to making research communication faster, fairer, and more useful. We do this by developing innovative software and high quality services for the global research community. Our growing team is made up of researchers and industry professionals working together to solve the most critical problems facing scientific publishing. Also discoverable on Platform About Our Team In Review Editorial Policies Advisory Board Help Center Resources Author Services Accessibility API Access RSS feed Manage Cookie Preferences © Research Square 2026 | ISSN 2693-5015 (online) Privacy Policy Terms of Service Do Not Sell My Personal Information {"props":{"pageProps":{"initialData":{"identity":"rs-4295510","acceptedTermsAndConditions":true,"allowDirectSubmit":true,"archivedVersions":[],"articleType":"Case Report","associatedPublications":[],"authors":[{"id":294703113,"identity":"1390e1d3-80f4-404c-b4bb-a81e3d321875","order_by":0,"name":"Yoshiki Fujikawa","email":"data:image/png;base64,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","orcid":"","institution":"Osaka Medical and Pharmaceutical University","correspondingAuthor":true,"prefix":"","firstName":"Yoshiki","middleName":"","lastName":"Fujikawa","suffix":""},{"id":294703114,"identity":"624aa4e9-56e4-49b0-804e-27ae42216199","order_by":1,"name":"Takahiro Fujishiro","email":"","orcid":"","institution":"Aoyama Neurosurgical Hospital (formerly Tanabe Neurosurgical Hospital)","correspondingAuthor":false,"prefix":"","firstName":"Takahiro","middleName":"","lastName":"Fujishiro","suffix":""},{"id":294703115,"identity":"17774c2b-6aea-49c4-80f8-8eb7e8a2f901","order_by":2,"name":"Hideki Tanabe","email":"","orcid":"","institution":"Aoyama Neurosurgical Hospital (formerly Tanabe Neurosurgical Hospital)","correspondingAuthor":false,"prefix":"","firstName":"Hideki","middleName":"","lastName":"Tanabe","suffix":""},{"id":294703116,"identity":"5be76128-ea9a-4749-b6e9-a6fea478c001","order_by":3,"name":"Toshihiro Takami","email":"","orcid":"","institution":"Osaka Medical and Pharmaceutical University","correspondingAuthor":false,"prefix":"","firstName":"Toshihiro","middleName":"","lastName":"Takami","suffix":""},{"id":294703117,"identity":"59d73da5-53a1-471b-bfe4-7d2a52304535","order_by":4,"name":"Masahiko Wanibuchi","email":"","orcid":"","institution":"Osaka Medical and Pharmaceutical University","correspondingAuthor":false,"prefix":"","firstName":"Masahiko","middleName":"","lastName":"Wanibuchi","suffix":""}],"badges":[],"createdAt":"2024-04-20 02:25:06","currentVersionCode":1,"declarations":"","doi":"10.21203/rs.3.rs-4295510/v1","doiUrl":"https://doi.org/10.21203/rs.3.rs-4295510/v1","draftVersion":[],"editorialEvents":[],"editorialNote":"","failedWorkflow":false,"files":[{"id":55525574,"identity":"c01b8c60-0f47-41d9-9750-f7a6a36c7c5f","added_by":"auto","created_at":"2024-04-29 14:43:44","extension":"png","order_by":1,"title":"Figure 1","display":"","copyAsset":false,"role":"figure","size":1720074,"visible":true,"origin":"","legend":"\u003cp\u003eComputed tomography (CT) images of intracranial chronic subdural hematoma (iCSDH). (A) Preoperative CT image shows left iCSDH with moderate midline shift. (B) Postoperative CT image shows that iCSDH is well evacuated.\u003c/p\u003e","description":"","filename":"Figure1.png","url":"https://assets-eu.researchsquare.com/files/rs-4295510/v1/b3b1a4bb697f44df712fd1a0.png"},{"id":55525579,"identity":"3c8d532f-7a53-45ba-8857-f889e6273d2c","added_by":"auto","created_at":"2024-04-29 14:43:44","extension":"png","order_by":2,"title":"Figure 2","display":"","copyAsset":false,"role":"figure","size":2265128,"visible":true,"origin":"","legend":"\u003cp\u003ePreoperative magnetic resonance imaging (MRI) of the spine. Both T1 (A and C) and T2 (B and D) hyperintense subdural hematomas (arrowheads in C and D) extend from the T11 to S2 levels (arrows in A and B), compressing the cauda equina. Cervical MRI displays no sign of hematoma (E: T1, and F: T2).\u003c/p\u003e","description":"","filename":"Figure2.png","url":"https://assets-eu.researchsquare.com/files/rs-4295510/v1/550565bdd6f058f17343093c.png"},{"id":55525580,"identity":"23b7c622-3101-45ac-bf26-41576d1f1876","added_by":"auto","created_at":"2024-04-29 14:43:44","extension":"png","order_by":3,"title":"Figure 3","display":"","copyAsset":false,"role":"figure","size":3586581,"visible":true,"origin":"","legend":"\u003cp\u003eIntraoperative photographs of spinal chronic subdural hematoma. (A) Liquefied dark-coloured hematoma is excreted after incision of the dura. (B) After wide opening of the dura, an outer membrane (arrow) of the hematoma is observed. (C) After rinsing the subdural hematoma, an intact arachnoid membrane (arrows) and clear cerebrospinal fluid are observed.\u003c/p\u003e","description":"","filename":"Figure3.png","url":"https://assets-eu.researchsquare.com/files/rs-4295510/v1/d9b1f3d1917775a8efcc17d0.png"},{"id":55526174,"identity":"eabf1cb1-128e-48e0-a3f7-3b18931b2760","added_by":"auto","created_at":"2024-04-29 14:51:44","extension":"png","order_by":4,"title":"Figure 4","display":"","copyAsset":false,"role":"figure","size":4361778,"visible":true,"origin":"","legend":"\u003cp\u003eThe histopathological images of the outer membrane of the spinal chronic subdural hematoma. The sections are stained by haematoxylin and eosin. Black dotted square (as shown in A) represents the area of the magnified image (B). The membrane comprises fibroblasts, collagen fibres, capillaries, macrophages, and hemosiderin (B).\u003c/p\u003e","description":"","filename":"Figure4.png","url":"https://assets-eu.researchsquare.com/files/rs-4295510/v1/39a41251c9685ee330acdca4.png"},{"id":55526173,"identity":"6924955d-19f9-4003-ad57-f3d6e0b821e4","added_by":"auto","created_at":"2024-04-29 14:51:44","extension":"png","order_by":5,"title":"Figure 5","display":"","copyAsset":false,"role":"figure","size":1492115,"visible":true,"origin":"","legend":"\u003cp\u003ePostoperative magnetic resonance imaging of spinal chronic subdural hematoma (sCSDH). After 4 months, no recurrence of sCSDH is observed (T1: A and C, T2: B and D).\u003c/p\u003e","description":"","filename":"Figure5.png","url":"https://assets-eu.researchsquare.com/files/rs-4295510/v1/c572cfdf94a0f54c2859135a.png"},{"id":55525575,"identity":"b72e0cfd-c5e4-49cb-9e59-8871f90e3dcb","added_by":"auto","created_at":"2024-04-29 14:43:44","extension":"png","order_by":6,"title":"Figure 6","display":"","copyAsset":false,"role":"figure","size":206150,"visible":true,"origin":"","legend":"\u003cp\u003ePossible pathogenesis of spinal chronic subdural hematoma (sCSDH). The first theory is direct trauma or spinal puncture to the lumbar area. This may cause disruption of small vessels such as bridging veins, resulting in sCSDH. The second theory is that the intracranial CSDH may migrate to the spine. The third theory is an increase in abdominal or thoracic pressure. This may also cause disruption of small vessels. The fourth theory is that the hematoma extends from the subarachnoid space to the subdural space. Black lines in the spinal canal represent the dura, while blue lines represent the arachnoid membrane. SDH, subdural hematoma.\u003c/p\u003e","description":"","filename":"Figure6.png","url":"https://assets-eu.researchsquare.com/files/rs-4295510/v1/3e8691b982d137d9d54939ec.png"},{"id":62887620,"identity":"f2f1eb2f-4fa9-4346-83d7-d29abba6c767","added_by":"auto","created_at":"2024-08-20 16:22:44","extension":"pdf","order_by":0,"title":"","display":"","copyAsset":false,"role":"manuscript-pdf","size":23745096,"visible":true,"origin":"","legend":"","description":"","filename":"manuscript.pdf","url":"https://assets-eu.researchsquare.com/files/rs-4295510/v1/c128e60b-6ae1-4d76-8cca-ffdc6d3eb344.pdf"}],"financialInterests":"No competing interests reported.","formattedTitle":"Pathogenesis of spinal chronic subdural hematoma based on histopathological findings: a case report","fulltext":[{"header":"Background","content":"\u003cp\u003eSubdural hematoma (SDH) often occurs in the intracranial space, whereas its occurrence along the spinal column in the extracranial space is rare [1, 2]. When presenting symptoms such as lower back pain, weakness in the lower extremities, or bowel and bladder dysfunction that persist for more than 2 weeks, it is classified as chronic SDH (CSDH) [3]. In the spine, the frequency of occurrence of CSDH is considerably lower than that of acute or subacute SDH [4].\u003c/p\u003e\n\u003cp\u003ePrevious studies have proposed various factors contributing to spinal CSDH (sCSDH), including a history of trauma, lumbar puncture, bleeding diathesis, use of antithrombotics, or vascular malformation, although instances of spontaneous occurrence have been noted [5]. In addition, various cases of concomitant intracranial CSDH (iCSDH) and sCSDH have been reported. However, the exact aetiology and pathogenesis of sCSDH remain unknown. In addition, few reports have considered histopathological findings in investigating the pathogenesis of sCSDH.\u003c/p\u003e\n\u003cp\u003eHerein, we present a case of concomitant iCSDH and sCSDH and discuss the pathogenesis of the disease based on the findings of histopathological examination.\u003c/p\u003e"},{"header":"Case Presentation","content":"\u003cp\u003e\u0026nbsp;A 76-year-old man, who had suffered from head trauma 1 month ago, was\u0026nbsp;transferred to our hospital after falling and sustaining head trauma again. He had a history of brain infarction a few decades prior to admission and had undergone burr hole surgery for a right iCSDH 4 years earlier. He was not on anticoagulant or antiplatelet medication. He presented with slight right hemiparesis (manual muscle test; MMT 4/5). Head computed tomography (CT) revealed a left iCSDH with a mild midline shift (Fig. 1A). He underwent burr hole surgery upon admission to our hospital, resulting in successful drainage of the hematoma (Fig. 1B). However, he remained unable to stand or walk unassisted 1 week after surgery. The MMT score for his legs was approximately 3 to 4, and his patellar tendon reflex was attenuated. The Babinski reflex was negative. Spinal magnetic resonance imaging (MRI) revealed a subdural hematoma extending from the T11 to S2 levels (Fig. 2A–D), with no evidence of hematoma at the cervical level (Fig. 2E, F).\u003c/p\u003e\n\u003cp\u003eTo improve his neurological condition, he underwent an L2–4 hemilaminectomy for hematoma evacuation. When the dura matter was opened, a dark-brownish liquid hematoma covered by the membrane resembling that of an iCSDH was observed (Fig. 3A, B). Subsequently the hematoma was washed and evacuated through saline irrigation using a drainage tube. Clear cerebrospinal fluid (CSF) was observed through the intact, normal arachnoid membrane, with no evidence of hemorrhage in the subarachnoid space (Fig. 3C).\u0026nbsp;\u003c/p\u003e\n\u003cp\u003eHistopathological examination revealed that the membrane of the hematoma contained fibroblasts, collagen fibres, capillaries, macrophages, and hemosiderin (Fig. 4A, B). Neither hemorrhage nor apparent cell infiltration was observed in the membrane.\u003c/p\u003e\n\u003cp\u003eThe patient was transferred to a rehabilitation hospital 9 days after lumbar surgery. Because of dementia, he lacked the willingness to rehabilitate, resulting in him being forced to use a wheelchair to move at discharge. Postoperative spinal MRI at 4 months showed no recurrence of subdural hematoma (Fig. 5A–D).\u003c/p\u003e"},{"header":"Discussion and Conclusions","content":"\u003cp\u003eIn this study, we report a rare case of concomitant iCSDH and sCSDH and the histopathological examination of its membrane. To the best of our knowledge, this is the first report to provide histopathological evidence of concomitant sCSDH and iCSDH.\u003c/p\u003e\n\u003cp\u003e\u0026nbsp;Intracranial SDH occurs spontaneously or is triggered predominantly by trauma [6-8], followed by intracranial hypotension [9]. Intracerebral hemorrhage [10, 11], ruptured cerebral aneurysm [12, 13], and cerebral vascular malformations [14-17] are occasionally accompanied by SDH, when bleeding extends to the subdural space. Other less common causes include vasculopathy [18] and neoplasm [19-21]. Development of iCSDH typically occurs as follows: after development of an acute SDH, the absorption of blood subsequently begins with breakdown of erythrocytes and other cellular components. Next, collagen synthesis is induced, and fibroblasts spread over the inner surface of the dura to form a thick outer membrane [22]. Subsequently, a thinner inner membrane forms, leading to the complete encapsulation of the clot. In general, this process spans approximately 2 weeks.\u003c/p\u003e\n\u003cp\u003e\u0026nbsp;The exact pathogenesis of sCSDH remains unknown (Fig. 6), with various theories proposed. One theory suggests that disruption of bridging veins in the spine, caused by direct trauma or lumbar puncture, leads to hematoma formation, similar to intracranial hematomas. The spinal subdural space, known to be less vascularised [23, 24], may contribute to the lower frequency of sCSDH compared to iCSDH. However, bridging veins exist not only in intracranial structures but also in the spine [25], suggesting that disruption of these veins can also cause a spinal SDH. A previous prospective study reported a significant association between direct impact to the lumbar area and spinal SDH in patients with iCSDH, suggesting double trauma to the head and lumbar regions as a major cause of concomitant intracranial and sCSDH. Another theory proposes that an iCSDH might migrate to the spinal area, supported by the less vascularised spinal subdural space and the subdural compartment between the cranium and spinal space proved to be connected [26]. Certain cases have shown hematomas in the posterior fossa or all through the spine [27, 28], with similar signal intensity on MRI, suggesting a common origin. However, our patient did not have a hematoma at the cervical level, as revealed through MRI. Furthermore, the previous prospective study found only 2 (1.2%) of 168 cases of iCSDH with concomitant spinal SDH on MRI [29]. A third theory suggests a different mechanism for the formation of sCSDH from that of iCSDH [30]. In this theory, sCSDH occurs due to a rapid increase in intravascular pressure of the intraspinal segments of lateral spinal vessels, caused by increased abdominal and thoracic pressure. This sudden increase in the intravascular pressure is not neutralised by a simultaneous increase in CSF pressure because of the shielding effect of the spinal column and its ligaments. Consequently, hemorrhage in the subdural space may occur. Such subdural accumulation of blood would be subject to the same set of forces that govern the formation of intracranial fluid collections. This theory is supported by a case report of sCSDH co-existing with a ligament flavum hematoma, indicating a mechanism consistent with this theory [31]. Finally, some researchers have stated that the subarachnoid hemorrhage is thought to extend into the subdural space [32, 33]. These potential pathogeneses may be more likely to be induced by the use of antithrombotics or because of bleeding diathesis.\u003c/p\u003e\n\u003cp\u003eDuring iCSDH formation, the outer and inner membranes that surround the hematoma form subsequently, as described previously [34]. According to histopathological examination, the outer membrane contains various components, such as neovasculature and fibrous tissue. Exudation from these neovasculatures contributes to hematoma enlargement. Previous studies have categorised histopathological features of the outer membrane into the following four subtypes: noninflammatory (I), inflammatory (II), hemorrhagic inflammatory (III), and scar inflammatory (IV) [35]. The noninflammatory (I) type comprises immature fibroblasts, collagen fibres, sparse cellular infiltration, and neocapillaries. The inflammatory (II) type comprises a single sheet of immature connective tissue with marked cellular infiltration and vascularisation. The hemorrhagic inflammatory (III) type consists of a few sheets of connective tissue with cellular infiltration and moderately-sized capillaries. The scar inflammatory (IV) type comprises inflammatory cell infiltration, neovascularisation, and hemorrhage. In addition, the outer membrane is thought to change from type I to IV in order [36]; thus, the histopathological appearance of the membrane is useful in estimating the duration of CSDH [37].\u0026nbsp;\u003c/p\u003e\n\u003cp\u003eIn our patient, the histopathological features of the outer membrane of the sCSDH were considered to be similar to those of type I iCSDH because of the slight cellular infiltration. Therefore, we suggest that in this case, the mechanism for sCSDH formation may be trauma to the lumbar area, similar to that for iCSDH resulting from head trauma, rather than migration. Moreover, based on the histopathology, this case was in the early phase of hematoma formation. Various studies have also reported that the outer membrane of the sCSDH was observed during surgery [4, 38], similar to our case. On the other hand, some studies did not find an outer membrane [39, 40], which may indicate that not enough time had elapsed for capsular formation or that the hematoma migrated from the cranial space.\u003c/p\u003e\n\u003cp\u003e\u0026nbsp;In conclusion, we demonstrated that sCSDH may have various potential pathogeneses, which can be elucidated through histopathological examination as in our case. This finding provides important insights for clinicians to enable them to correctly diagnose and appropriately manage sCSDH.\u003c/p\u003e"},{"header":"Abbreviations","content":"\u003cp\u003eCSDH, chronic subdural hematoma\u003c/p\u003e\n\u003cp\u003eCSF, cerebrospinal fluid\u003c/p\u003e\n\u003cp\u003eCT, computed tomography\u003c/p\u003e\n\u003cp\u003eiCSDH, intracranial chronic subdural hematoma\u003c/p\u003e\n\u003cp\u003eMMT, manual muscle test\u003c/p\u003e\n\u003cp\u003eMRI, magnetic resonance imaging\u003c/p\u003e\n\u003cp\u003esCSDH, spinal chronic subdural hematoma\u003c/p\u003e\n\u003cp\u003eSDH, Subdural hematoma\u003c/p\u003e"},{"header":"Declarations","content":"\u003ch2\u003eEthics approval and consent to participate\u003c/h2\u003e\n\u003cp\u003eApproval from an ethics review committee was not sought as the publication is a case report.\u003c/p\u003e\n\u003ch2\u003eConsent for publication\u003c/h2\u003e\n\u003cp\u003eWe obtained written informed consent for publication from the patient.\u003c/p\u003e\n\u003ch2\u003eAvailability of data and materials\u003c/h2\u003e\n\u003cp\u003eNot applicable.\u003c/p\u003e\n\u003ch2\u003eCompeting interests\u003c/h2\u003e\n\u003cp\u003eThe authors declare that they have no competing interests.\u003c/p\u003e\n\u003ch2\u003eFunding\u003c/h2\u003e\n\u003cp\u003eNone.\u003c/p\u003e\n\u003ch2\u003eAuthors’ contributions\u003c/h2\u003e\n\u003cp\u003eYF, TF, and HT were involved in diagnosis and managing the patient. YF drafted the manuscript while TT critically revised it. MW supervised this study. All authors read and approved the final manuscript.\u003c/p\u003e\n\u003ch2\u003eAcknowledgements\u003c/h2\u003e\n\u003cp\u003eThe authors would like to acknowledge Prof. Seiichi Hirota (Department of Surgical Pathology, Hyogo College of Medicine, Nishinomiya, Hyogo, Japan) for his advice regarding histopathological analysis. We also would like to thank Aya Sunamura and Akemi Usui for their secretarial work and Editage for English language editing.\u003c/p\u003e"},{"header":"References","content":"\u003col\u003e\n\u003cli\u003ede Beer MH, Eysink Smeets MM, Koppen H. Spontaneous Spinal Subdural Hematoma. Neurologist. 2017;22 1:34-9; doi: 10.1097/nrl.0000000000000100.\u003c/li\u003e\n\u003cli\u003eBenyaich Z, Laghmari M, Lmejjati M, Aniba K, Ghannane H, Ait Benali S. Acute Lumbar Spinal Subdural Hematoma Inducing Paraplegia After Lumbar Spinal Manipulation: Case Report and Literature Review. 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United States; 2020. p. 703-5.\u003c/li\u003e\n\u003cli\u003eStone R, Jones O: Encephalography: a Review of 113 cases, and a Report of Postmortem Studies on the Injection of Air. In., vol. 21: Radiology; 1933: 411-9.\u003c/li\u003e\n\u003cli\u003eJain V, Singh J, Sharma R. Spontaneous concomitant cranial and spinal subdural haematomas with spontaneous resolution. Singapore Med J. 2008;49 2:e53-8.\u003c/li\u003e\n\u003cli\u003eLecouvet FE, Annet L, Duprez TP, Cosnard G, Scordidis V, Malghem J. Uncommon magnetic resonance imaging observation of lumbar subdural hematoma with cranial origin. J Comput Assist Tomogr. 2003;27 4:530-3; doi: 10.1097/00004728-200307000-00013.\u003c/li\u003e\n\u003cli\u003eKokubo R, Kim K, Mishina M, Isu T, Kobayashi S, Yoshida D, et al. Prospective assessment of concomitant lumbar and chronic subdural hematoma: is migration from the intracranial space involved in their manifestation? 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World Neurosurg. 2017;108:954-8; doi: 10.1016/j.wneu.2017.09.063.\u003c/li\u003e\n\u003cli\u003eNagahori T, Nishijima M, Takaku A. [Histological study of the outer membrane of chronic subdural hematoma: possible mechanism for expansion of hematoma cavity]. No Shinkei Geka. 1993;21 8:697-701.\u003c/li\u003e\n\u003cli\u003eSato S, Suzuki J. Ultrastructural observations of the capsule of chronic subdural hematoma in various clinical stages. J Neurosurg. 1975;43 5:569-78; doi: 10.3171/jns.1975.43.5.0569.\u003c/li\u003e\n\u003cli\u003eBokka S, Trivedi A. Histopathological study of the outer membrane of the dura mater in chronic sub dural hematoma: Its clinical and radiological correlation. Asian J Neurosurg. 2016;11 1:34-8; doi: 10.4103/1793-5482.154979.\u003c/li\u003e\n\u003cli\u003eLeber KA, Pendl G, Kogler S, Kammerhuber F, Ebner F. Simultaneous spinal and intracranial chronic subdural hematoma. Case illustration. J Neurosurg. 1997;87 4:644; doi: 10.3171/jns.1997.87.4.0644.\u003c/li\u003e\n\u003cli\u003eKanamaru H, Kanamaru K, Araki T, Hamada K. Simultaneous Spinal and Intracranial Chronic Subdural Hematoma Cured by Craniotomy and Laminectomy: A Video Case Report. In: Case Rep Neurol. Switzerland; 2016. p. 72-7.\u003c/li\u003e\n\u003cli\u003eMatsumoto H, Matsumoto S, Yoshida Y. Concomitant Intracranial Chronic Subdural Hematoma and Spinal Subdural Hematoma: A Case Report and Literature Review. World Neurosurg. 2016;90:706.e1-.e9; doi: 10.1016/j.wneu.2016.03.020.\u003c/li\u003e\n\u003c/ol\u003e"}],"fulltextSource":"","fullText":"","funders":[],"hasAdminPriorityOnWorkflow":false,"hasManuscriptDocX":true,"hasOptedInToPreprint":true,"hasPassedJournalQc":"","hasAnyPriority":false,"hideJournal":true,"highlight":"","institution":"","isAcceptedByJournal":false,"isAuthorSuppliedPdf":false,"isDeskRejected":"","isHiddenFromSearch":false,"isInQc":false,"isInWorkflow":false,"isPdf":false,"isPdfUpToDate":true,"isWithdrawnOrRetracted":false,"journal":{"display":true,"email":"[email protected]","identity":"researchsquare","isNatureJournal":false,"hasQc":true,"allowDirectSubmit":true,"externalIdentity":"","sideBox":"","snPcode":"","submissionUrl":"/submission","title":"Research Square","twitterHandle":"researchsquare","acdcEnabled":true,"dfaEnabled":false,"editorialSystem":"","reportingPortfolio":"","inReviewEnabled":false,"inReviewRevisionsEnabled":true},"keywords":"Chronic subdural hematoma, Histopathology, Pathogenesis, Spinal subdural hematoma","lastPublishedDoi":"10.21203/rs.3.rs-4295510/v1","lastPublishedDoiUrl":"https://doi.org/10.21203/rs.3.rs-4295510/v1","license":{"name":"CC BY 4.0","url":"https://creativecommons.org/licenses/by/4.0/"},"manuscriptAbstract":"\u003cp\u003eBackground\u003c/p\u003e\n\u003cp\u003eSpinal chronic subdural hematoma (sCSDH) is a rare condition whereas intracranial chronic subdural hematoma (iCSDH) is well-recognised and documented in clinical settings. Despite various theories have been proposed, the exact pathogenesis of sCSDH remains to be elucidated. Herein, we report a rare case of sCSDH with co-existing iCSDH and deduce its etiology using histopathological findings.\u003c/p\u003e\n\u003cp\u003eCase presentation\u003c/p\u003e\n\u003cp\u003eA 76-year-old Japanese man with slight hemiparesis due to iCSDH underwent burr hole surgery with successful drainage of the hematoma. The patient could not walk unassisted after the surgery. Spinal magnetic resonance imaging (MRI) was performed, revealing a subdural hematoma extending from the T11 to S2 levels. The patient decided to undergo hematoma evacuation via a hemilaminectomy. Intraoperatively, we identified a dark liquefied hematoma with its capsule, which closely resembled an iCSDH. Histological examination of the resected capsule revealed loose fibrovascular tissues comprising capillaries, collagen fibres, small number of macrophages, and hemosiderin deposits. These findings were very similar to those for iCSDH, such that the pathogenesis of sCSDH was considered identical to that of iCSDH. The patient was transferred to a rehabilitation hospital, and 4 months after the lumbar surgery, no recurrence was observed in MRI examination.\u003c/p\u003e\n\u003cp\u003eConclusions\u003c/p\u003e\n\u003cp\u003esCSDH may result from various potential pathogeneses, that clinicians should consider for correct diagnosis and appropriate management. Our case provides valuable insights into the pathogenesis of sCSDH.\u003c/p\u003e","manuscriptTitle":"Pathogenesis of spinal chronic subdural hematoma based on histopathological findings: a case report","msid":"","msnumber":"","nonDraftVersions":[{"code":1,"date":"2024-04-29 14:43:39","doi":"10.21203/rs.3.rs-4295510/v1","editorialEvents":[{"type":"communityComments","content":0}],"status":"published","journal":{"display":true,"email":"[email protected]","identity":"researchsquare","isNatureJournal":false,"hasQc":true,"allowDirectSubmit":true,"externalIdentity":"","sideBox":"","snPcode":"","submissionUrl":"/submission","title":"Research Square","twitterHandle":"researchsquare","acdcEnabled":true,"dfaEnabled":false,"editorialSystem":"","reportingPortfolio":"","inReviewEnabled":false,"inReviewRevisionsEnabled":true}}],"origin":"","ownerIdentity":"a44c1f0e-028b-4539-882d-5c9a06ab57c7","owner":[],"postedDate":"April 29th, 2024","published":true,"recentEditorialEvents":[],"rejectedJournal":[],"revision":"","amendment":"","status":"posted","subjectAreas":[],"tags":[],"updatedAt":"2024-08-20T16:14:26+00:00","versionOfRecord":[],"versionCreatedAt":"2024-04-29 14:43:39","video":"","vorDoi":"","vorDoiUrl":"","workflowStages":[]},"version":"v1","identity":"rs-4295510","journalConfig":"researchsquare"},"__N_SSP":true},"page":"/article/[identity]/[[...version]]","query":{"redirect":"/article/rs-4295510","identity":"rs-4295510","version":["v1"]},"buildId":"qtupq5eGEP_6zYnWcrvyt","isFallback":false,"isExperimentalCompile":false,"dynamicIds":[84888],"gssp":true,"scriptLoader":[]}