Hydatid Disease Contributes to Spinal Cord Compression: A Case Report and Literature Review

Hydatid Disease Contributes to Spinal Cord Compression: A Case Report and Literature Review | 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 Hydatid Disease Contributes to Spinal Cord Compression: A Case Report and Literature Review Jinchao Cheng, Bing Ma, Min Li, Kai Zhao, Haifeng Li, Ruijun Bai This is a preprint; it has not been peer reviewed by a journal. https://doi.org/ 10.21203/rs.3.rs-4914019/v1 This work is licensed under a CC BY 4.0 License Status: Under Review Version 1 posted 11 You are reading this latest preprint version Abstract Background Hydatid disease is an infectious disease caused by echinococcus that causes liver and lung damage in humans. Bone involvement is rare and accounts for only 0.5%-4% of hydatid diseases; the spine is more prone to be infected with a proportion of 50% in musculoskeletal tissue. Missed hydatid cysts may lead to serious consequence, it is necessary to diagnosis and treatment of hydatid cysts at early stage. Case presentation: We present a case of a 47yearold Han Chinese male diagnosed with spinal hydatidosis. The patient lived in pasturing areas accompanied by symptoms of low back pain radiating to the legs, progressive weakness, and hypoesthesia in the lower limbs accompanied by difficulty walking. Computed tomography (CT) images showed a spinal hydatid cyst in the lumbar vertebra2 (L2) that caused bone destruction in the vertebral body and lamina. Magnetic resonance imaging (MRI) images showed hyperintense cystic components on L2 with the appearance of a lobulated, multiocular, septate cystic mass and compression of the spinal cord at the segments of L2. The patient underwent subtotal vertebral resection to completely remove the damaged vertebral and paravertebral cysts, and histopathological examination revealed the characteristic features of hydatid cysts. The patient received a postoperative antiparasitic drug treatment and finally achieved a successful treatment. Conclusions Spinal hydatidosis is a rare infectious disease. It should be considered as an important differential diagnosis for patients accompanying by spinal bone destruction with animal contact history. Surgical removal combined with antiviral drugs is an effective way to treat spinal hydatidosis. In addition, long term follow-up is mandatory for avoiding its recurrence. Hydatid disease Spine MRI imaging Surgery Therapy Figures Figure 1 Figure 2 Introduction Hydatidosis is a rare infection caused by echinococcus and mainly spread throughout the Mediterranean Sea, Asia, North and East Africa, South America, Australia, and Middle East [ 1 , 2 ]. It can affect any organ of the body, and the most commonly infected organs are liver and lungs [ 3 ]. The incidence of echinococcosis in musculoskeletal system was low, accounting for 0.5%-4% of all case [ 4 ]. Approximately 50% of bone hydatidosis cases are spinal, and most cases of spinal hydatidosis involve the thoracic spine [ 5 , 6 ]. Due to the low incidence and special infection site in spinal hydatidosis, diagnosis is difficult in clinics and radiography. Herein, we report a case of a 47-year-old man with a spinal-paraspinal hydatid cyst that caused spinal cord compression and vertebral column destruction. The patient was successfully treated with total surgical excision and pharmaceutical treatment. We discuss the symptoms and differential diagnosis of this case and review the relevant literature. Case Report A 47-year-old Han Chinese male patient presented with a 5-month history of low back pain radiating to the legs, progressive weakness, and hypoesthesia in the lower limbs, accompanied by difficulty walking for 1 month. The patient lived in a pasturing area and worked primarily on herding animals with no history of fever or tuberculosis. Physical examination revealed a normal tone in both lower limbs. Power examination revealed grade 4 in the right lower limb, accompanied by grade 4 proximal and grade 3 distal to the left lower limb. Initial laboratory examination results showed that the patient had normal laboratory findings, including complete blood cell count, serum chemistry, and erythrocyte sedimentation rate testing; C-reactive protein (CRP) was 7.93 mg/dL, carcinoembryonic antigen (CEA) was 3.84ng/ml, and a test for mycobacterium tuberculosis was positive. Imaging examinations include radiography, computed tomography (CT), and magnetic resonance imaging (MRI). Radiography revealed a bone defect in lumbar2 (L2) with the side effect of decreasing the height of vertebral body (Figure-1A and B). Three-dimensional CT scans showed osteolytic destruction of L2 (Figure-1C), and sagittal CT images confirmed osteolytic destruction of L2 and posterior spinous process (Figure-1D). Axial imaging showed the bone destruction of L2 was mainly concentrated in the left posterior part of vertebral body. The left pedicle and lamina were also affected and resulted in bone loss (Figure-1E). As previous examinations showed elevated CEA levels and osteolytic destruction in vertebra, we performed whole-body emission computed tomography (ECT) to exclude the disease of osteosarcoma (Figure-1F). The magnetic resonance imaging (MRI) images revealed multiple cystic components in the sagittal plane of the L2. It presented lobulated, multi-eyed, and honeycomb appearance partially existing in the spinal canal at the posterior edge of L2 in T1-weighted scans (Figure-1G, H), T2-weighted sagittal images showed high intensity cystic component images of L2 and its posterior spinal canal, and multiple scattered large cystic block images were also seen at the posterior margin of lumbar vertebrae1 body with clear boundary between each other accompanied with obvious compression of the dural sac at the corresponding site (Figure-1I, J). We performed cyst removal through posterior cystectomy and L1-L2 spinal joint fusion under general anesthesia. In general, the patient underwent a posterior midline approach to remove the lesions at the posterior edge of the L1 and L2 vertebral bodies, subtotal L2 vertebral resection and lumbar 1–2 discectomy, subsequently L2 was replaced with a mesh cage. Intraoperative cystic lesions had a white crystal-like appearance (Figure-2A), with white granular tissue infiltrating the entire vertebral body and canal, thus disrupting the integrity of the vertebral body. The pathological results showed a large number of inflammatory cells and macrophage invasion in the vertebral body, consistent with the pathological damage caused by hydatid disease (Figure-2B). Before the end of the operation, the surgical site was irrigated with a hypertonic sodium chloride solution to prevent hydatid recurrence. The patient was postoperatively treated with pharmacotherapy. During the treatment period, hematopoietic and liver enzyme changes were monitored according to the treatment guidelines. The patient had a good recovery state of both lower limb powers, along with no signs of local recurrence at the latest examination one year postoperatively. Discussion Spinal hydatidosis is a rare disease which caused bone infection in adults. Because spinal hydatidosis has no special characteristic signs or symptoms, it is difficult to diagnose first and may be misdiagnosed as other common diseases such as lumbar intervertebral protrusion and lumbar spinal stenosis. Previous reports have shown that spinal hydatidosis occurs in 60% in thoracic vertebrae, 35% in lumbosacral vertebrae, and 5% in cervical vertebrae respectively [ 7 ]. The clinical symptoms of vertebral echinococcosis mainly include lumbar pain (59.2%-75%) accompanied by lower extremity weakness and paresthesia (37%-50%) [ 8 , 9 ]. In the present case, lower back pain and decreased power in both lower limbs accompanied paresthesia. We first misdiagnosed the patient with bone tumors or tuberculosis because of an elevated CEA level, a positive examination for tuberculosis, and vertebral body destruction. Due to the absence of signs on ECT scans and the patient history of animal herding in pastoral areas combined with the MRI images, we temporarily excluded bone tumors and considered the possibility of echinococcosis infection. The spinal cystic echinococcosis showed no characteristic signs or symptoms. Therefore, these cases are rarely detected and diagnosed in the early stages, until symptoms of vertebral destruction and compression of the spinal cord and nerve caused by enlarged cysts appear. Echinococcus diagnosis should always be considered the diagnosis of echinococcus for patients living in rural areas, especially in those in contact with animals. Previous studies have shown that spinal hydatid disease is mainly caused by echinococcus granulosus, which leads to its cystic form, resulting in spinal pathological changes [ 10 ]. Computed tomography (CT) and magnetic resonance imaging (MRI) are the primary imaging modalities used to diagnose spinal hydatid disease. According to the CT and MRI findings, spinal hydatid can be divided into five radiological types: intramedullary, intradural extramedullary, epidural, vertebral echinococcus, and paravertebral lesions extending into spinal structures [ 11 ]. Among the five radiological types, vertebral echinococcus and paravertebral lesions extending into the spinal structures are more common and are seen in the majority of patients [ 11 ]. Zhang et al. [ 12 ] found the MRI feature of spinal hydatid disease presented as a “bunch of grapes” was a typical imaging and the total vertebral resection had a better therapeutic effect than subtotal vertebral resection. The imaging examination in our patient showed that the lesions of spinal hydatid were mainly located in L2. Moreover, the MRI images of the patient we reported to show polycystic lobulated clusters of grape beads, which is consistent with previous reports [ 12 ]. The theoretical cure for spinal hydatidosis is resection of the lesion and a preference for recurrence, but the cure rate is low when surgical intervention is performed alone. The main goal of surgical intervention for spinal echinococcosis is to completely remove the damaged part of the vertebral body and achieve healthy vertebral margins on both sides of the resection [ 13 ]. Zhang et al. [ 12 ] found the total vertebrotomy combined with achieved antiparasitic drug therapy achieved a better therapeutic effect, moreover they found antiparasitic drug play an important role in preventing recurrence. Some studies have also reported that preoperative combined with postoperative antiparasitic drugs is an important adjuvant treatment for its recurrence [ 6 , 12 ]. In our case, we found that subtotal vertebral resection could completely remove hydatid cysts without any residue. During surgery, we carefully and integrally removed the cyst and rinsed and soaked the wound with hypertonic saline to prevent recurrence. However, it is still unclear whether recurrence can be prevented. Subsequently, the patient was treated with antiparasitic drug therapy as an important supplementary therapy to prevent recurrence. The patient recovered well and had normal power in both lower limbs without low back pain at the last follow-up. Conclusion We report a rare case of typical spinal hydatidosis treated by subtotal vertebral resection combined use of antiparasitic drugs and achieved a good therapeutic effect. Surgical removal combined with antiviral drugs is an effective treatment for spinal hydatidosis. Abbreviations CT computed tomography MRI magnetic resonance imaging CRP C-reactive protein CEA carcinoembryonic antigen L2 lumbar2 ECT emission computed tomography. Declarations We declared that all the methods and clinical data in this study was approved by the medical ethical committee at Second Hospital of Lanzhou University (2023A—160). All the authors provided written informed consent to participate in this study. Consent for publication Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal. Conflicts of interest The authors declare that there are no conflicts of interest. Ethical approval The medical ethical committee at Second Hospital of Lanzhou University (No. 2023A—160). Supplementary data None. Funding This study was supported by Key R&D Plan of Gansu Provincial Department of Science and Technology-Social Development Projects (Grant No. 23YFFA0043) and the Natural Science Foundation of Wuxi City (No. k20231060). Author Contribution Ruijun Bai and Min Li conceived the project. Jinchao Cheng and Ruijun Bai drafted the manuscript. Ruijun Bai and Min Li performed the statistical analyses. Jinchao Cheng, Bing Ma, Kai Zhao, Haifeng Li revised the manuscript. All authors reviewed the manuscript. All authors read and approved the final manuscript. Acknowledgement We thank the patient and members of the team. Data Availability The data analyzed in the study are available from the corresponding author upon reasonable request. References Annamalai S, Muthu S, Thakur A, Ramakrishnan E. Novel Multimodal Treatment Regimen for the Management of Primary Sacrococcygeal Cystic Echinococcosis. J Orthop Case Rep. 2021;11 11:22–6; doi: 10.13107/jocr.2021.v11.i11.2500 . Grosso G, Gruttadauria S, Biondi A, Marventano S, Mistretta A. Worldwide epidemiology of liver hydatidosis including the Mediterranean area. World J Gastroenterol. 2012;18 13:1425–37; doi: 10.3748/wjg.v18.i13.1425 . Ozdemir HM, Ogun TC, Tasbas B. A lasting solution is hard to achieve in primary hydatid disease of the spine: long-term results and an overview. Spine (Phila Pa 1976). 2004;29 8:932–7; doi: 10.1097/00007632-200404150-00022 . Al-Mendalawi MD. Hydatid disease of the spine: A rare case. J Craniovertebr Junction Spine. 2017;8 3:291; doi: 10.4103/jcvjs.JCVJS_71_17 . Govender TS, Aslam M, Parbhoo A, Corr P. Hydatid disease of the spine. A long-term followup after surgical treatment. Clin Orthop Relat Res. 2000; 378:143–7; doi: 10.1097/00003086-200009000-00023 . Liang Q, Xiang H, Xu L, Wen H, Tian Z, Yunus A, et al. Treatment experiences of thoracic spinal hydatidosis: a single-center case-series study. Int J Infect Dis. 2019;89:163–8; doi: 10.1016/j.ijid.2019.09.024 . Cavus G, Acik V, Bilgin E, Gezercan Y, Okten AI. Endless story of a spinal column hydatid cyst disease: A case report. Acta Orthop Traumatol Turc. 2018;52 5:397–403; doi: 10.1016/j.aott.2018.03.004 . Gezercan Y, Okten AI, Cavus G, Acik V, Bilgin E. Spinal Hydatid Cyst Disease. World Neurosurg. 2017;108:407–17; doi: 10.1016/j.wneu.2017.09.015 . Monge-Maillo B, Olmedo Samperio M, Perez-Molina JA, Norman F, Mejia CR, Tojeiro SC, et al. Osseous cystic echinococcosis: A case series study at a referral unit in Spain. PLoS Negl Trop Dis. 2019;13 2:e0007006; doi: 10.1371/journal.pntd.0007006 . Gennari A, Almairac F, Litrico S, Albert C, Marty P, Paquis P. Spinal cord compression due to a primary vertebral hydatid disease: A rare case report in metropolitan France and a literature review. Neurochirurgie. 2016;62 4:226–8; doi: 10.1016/j.neuchi.2016.03.001 . Braithwaite PA, Lees RF. Vertebral hydatid disease: radiological assessment. Radiology. 1981;140 3:763–6; doi: 10.1148/radiology.140.3.7280247 . Zhang T, Ma LH, Liu H, Li SK. Incurable and refractory spinal cystic echinococcosis: A case report. World J Clin Cases. 2021;9 33:10337–44; doi: 10.12998/wjcc.v9.i33.10337 . Caglar YS, Ozgural O, Zaimoglu M, Kilinc C, Eroglu U, Dogan I, et al. Spinal Hydatid Cyst Disease: Challenging Surgery - an Institutional Experience. J Korean Neurosurg Soc. 2019;62 2:209–16; doi: 10.3340/jkns.2017.0245 . Additional Declarations No competing interests reported. Cite Share Download PDF Status: Under Review Version 1 posted Editorial decision: Revision requested 16 Oct, 2024 Reviewers agreed at journal 30 Sep, 2024 Reviews received at journal 27 Sep, 2024 Reviewers agreed at journal 25 Sep, 2024 Reviewers agreed at journal 09 Sep, 2024 Reviews received at journal 07 Sep, 2024 Reviewers agreed at journal 02 Sep, 2024 Reviewers invited by journal 28 Aug, 2024 Editor assigned by journal 28 Aug, 2024 Submission checks completed at journal 28 Aug, 2024 First submitted to journal 14 Aug, 2024 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. 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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-4914019","acceptedTermsAndConditions":true,"allowDirectSubmit":false,"archivedVersions":[],"articleType":"Case Report","associatedPublications":[],"authors":[{"id":358755765,"identity":"402b04e5-56fb-471b-91c2-7bf8d8019530","order_by":0,"name":"Jinchao Cheng","email":"","orcid":"","institution":"Xuancheng Central Hospital","correspondingAuthor":false,"prefix":"","firstName":"Jinchao","middleName":"","lastName":"Cheng","suffix":""},{"id":358755766,"identity":"ddeca1a7-1265-44c4-8497-ad0eac02fb78","order_by":1,"name":"Bing Ma","email":"","orcid":"","institution":"Second Hospital of Lanzhou University","correspondingAuthor":false,"prefix":"","firstName":"Bing","middleName":"","lastName":"Ma","suffix":""},{"id":358755767,"identity":"5285d3c5-41ba-4ea2-b298-b828e6e733ac","order_by":2,"name":"Min Li","email":"","orcid":"","institution":"Soochow University","correspondingAuthor":false,"prefix":"","firstName":"Min","middleName":"","lastName":"Li","suffix":""},{"id":358755768,"identity":"3ecf2783-0cde-4708-be28-708edec0f9b8","order_by":3,"name":"Kai Zhao","email":"","orcid":"","institution":"Soochow University","correspondingAuthor":false,"prefix":"","firstName":"Kai","middleName":"","lastName":"Zhao","suffix":""},{"id":358755772,"identity":"8af4047e-12f7-42d3-9dd8-712a60ad157a","order_by":4,"name":"Haifeng Li","email":"","orcid":"","institution":"Soochow University","correspondingAuthor":false,"prefix":"","firstName":"Haifeng","middleName":"","lastName":"Li","suffix":""},{"id":358755773,"identity":"52d3bc8b-a8df-4df9-b51d-53106430ea59","order_by":5,"name":"Ruijun Bai","email":"data:image/png;base64,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","orcid":"","institution":"Soochow University","correspondingAuthor":true,"prefix":"","firstName":"Ruijun","middleName":"","lastName":"Bai","suffix":""}],"badges":[],"createdAt":"2024-08-14 13:33:23","currentVersionCode":1,"declarations":"","doi":"10.21203/rs.3.rs-4914019/v1","doiUrl":"https://doi.org/10.21203/rs.3.rs-4914019/v1","draftVersion":[],"editorialEvents":[],"editorialNote":"","failedWorkflow":false,"files":[{"id":66904909,"identity":"3e3e21f5-8bd4-43d7-bbb3-1825366d65a2","added_by":"auto","created_at":"2024-10-17 17:47:04","extension":"png","order_by":1,"title":"Figure 1","display":"","copyAsset":false,"role":"figure","size":11254169,"visible":true,"origin":"","legend":"\u003cp\u003e\u003cstrong\u003eRadiology images and ECT scans of the spine.\u003c/strong\u003e(A) A sagittal view of the radiographic images shows bone destruction and cavity formation at the posterior edge of L2. (B) Front-to-back view of radiographic images showing decreased mineral density of L2 accompanied by cavity formation. (C)\u003cstrong\u003e \u003c/strong\u003eFront-to-back view of CT images showed the destruction of L2. (D) Sagittal view of CT images showed bone destruction and cavity formation at the center of L2 and the pedicle was also involved. (E) Axial view of CT images showed bone destruction mainly at the posterior left side of the vertebra along with partial defect of pedicle. (F) ECT scanning found there presented no hyperintense images in the whole body.\u003cstrong\u003e \u003c/strong\u003e(G, H)\u003cstrong\u003e \u003c/strong\u003eT1-weighted image showed heterogeneous low signal intensity and clear boundary in L2 and the paraspinal region; (I, J)\u003cstrong\u003e \u003c/strong\u003eT2-weighted images reveal multiple areas of hyperintense lesion and spinal cord compression.\u003c/p\u003e","description":"","filename":"Fig1.png","url":"https://assets-eu.researchsquare.com/files/rs-4914019/v1/b0e72f1ef0849f954da42f43.png"},{"id":66904908,"identity":"f244153b-2946-47df-a5a7-5c6443aec506","added_by":"auto","created_at":"2024-10-17 17:47:04","extension":"png","order_by":2,"title":"Figure 2","display":"","copyAsset":false,"role":"figure","size":4462163,"visible":true,"origin":"","legend":"\u003cp\u003e\u003cstrong\u003eThe grossappearance and pathological imagesof the cysts after operation. \u003c/strong\u003e(A) The gross appearance after surgery were consistent with the standard of hydatid cysts, with partial cysts and thick cyst walls. (B) The pathological images showed the cysts and vacuoles consistent with hydatid disease.\u003c/p\u003e","description":"","filename":"Fig2.png","url":"https://assets-eu.researchsquare.com/files/rs-4914019/v1/2dcdd36ab03853e9eea4fca0.png"},{"id":66905449,"identity":"00266110-436c-4fea-8583-02377595a604","added_by":"auto","created_at":"2024-10-17 17:55:11","extension":"pdf","order_by":0,"title":"","display":"","copyAsset":false,"role":"manuscript-pdf","size":14709630,"visible":true,"origin":"","legend":"","description":"","filename":"manuscript.pdf","url":"https://assets-eu.researchsquare.com/files/rs-4914019/v1/a612dc0f-c19d-4501-b582-879997c1a3c8.pdf"}],"financialInterests":"No competing interests reported.","formattedTitle":"Hydatid Disease Contributes to Spinal Cord Compression: A Case Report and Literature Review","fulltext":[{"header":"Introduction","content":"\u003cp\u003eHydatidosis is a rare infection caused by echinococcus and mainly spread throughout the Mediterranean Sea, Asia, North and East Africa, South America, Australia, and Middle East [\u003cspan citationid=\"CR1\" class=\"CitationRef\"\u003e1\u003c/span\u003e, \u003cspan citationid=\"CR2\" class=\"CitationRef\"\u003e2\u003c/span\u003e]. It can affect any organ of the body, and the most commonly infected organs are liver and lungs [\u003cspan citationid=\"CR3\" class=\"CitationRef\"\u003e3\u003c/span\u003e]. The incidence of echinococcosis in musculoskeletal system was low, accounting for 0.5%-4% of all case [\u003cspan citationid=\"CR4\" class=\"CitationRef\"\u003e4\u003c/span\u003e]. Approximately 50% of bone hydatidosis cases are spinal, and most cases of spinal hydatidosis involve the thoracic spine [\u003cspan citationid=\"CR5\" class=\"CitationRef\"\u003e5\u003c/span\u003e, \u003cspan citationid=\"CR6\" class=\"CitationRef\"\u003e6\u003c/span\u003e]. Due to the low incidence and special infection site in spinal hydatidosis, diagnosis is difficult in clinics and radiography. Herein, we report a case of a 47-year-old man with a spinal-paraspinal hydatid cyst that caused spinal cord compression and vertebral column destruction. The patient was successfully treated with total surgical excision and pharmaceutical treatment. We discuss the symptoms and differential diagnosis of this case and review the relevant literature.\u003c/p\u003e"},{"header":"Case Report","content":"\u003cp\u003eA 47-year-old Han Chinese male patient presented with a 5-month history of low back pain radiating to the legs, progressive weakness, and hypoesthesia in the lower limbs, accompanied by difficulty walking for 1 month. The patient lived in a pasturing area and worked primarily on herding animals with no history of fever or tuberculosis.\u003c/p\u003e \u003cp\u003ePhysical examination revealed a normal tone in both lower limbs. Power examination revealed grade 4 in the right lower limb, accompanied by grade 4 proximal and grade 3 distal to the left lower limb.\u003c/p\u003e \u003cp\u003eInitial laboratory examination results showed that the patient had normal laboratory findings, including complete blood cell count, serum chemistry, and erythrocyte sedimentation rate testing; C-reactive protein (CRP) was 7.93 mg/dL, carcinoembryonic antigen (CEA) was 3.84ng/ml, and a test for mycobacterium tuberculosis was positive.\u003c/p\u003e \u003cp\u003eImaging examinations include radiography, computed tomography (CT), and magnetic resonance imaging (MRI). Radiography revealed a bone defect in lumbar2 (L2) with the side effect of decreasing the height of vertebral body (Figure-1A and B). Three-dimensional CT scans showed osteolytic destruction of L2 (Figure-1C), and sagittal CT images confirmed osteolytic destruction of L2 and posterior spinous process (Figure-1D). Axial imaging showed the bone destruction of L2 was mainly concentrated in the left posterior part of vertebral body. The left pedicle and lamina were also affected and resulted in bone loss (Figure-1E). As previous examinations showed elevated CEA levels and osteolytic destruction in vertebra, we performed whole-body emission computed tomography (ECT) to exclude the disease of osteosarcoma (Figure-1F). The magnetic resonance imaging (MRI) images revealed multiple cystic components in the sagittal plane of the L2. It presented lobulated, multi-eyed, and honeycomb appearance partially existing in the spinal canal at the posterior edge of L2 in T1-weighted scans (Figure-1G, H), T2-weighted sagittal images showed high intensity cystic component images of L2 and its posterior spinal canal, and multiple scattered large cystic block images were also seen at the posterior margin of lumbar vertebrae1 body with clear boundary between each other accompanied with obvious compression of the dural sac at the corresponding site (Figure-1I, J).\u003c/p\u003e \u003cp\u003eWe performed cyst removal through posterior cystectomy and L1-L2 spinal joint fusion under general anesthesia. In general, the patient underwent a posterior midline approach to remove the lesions at the posterior edge of the L1 and L2 vertebral bodies, subtotal L2 vertebral resection and lumbar 1\u0026ndash;2 discectomy, subsequently L2 was replaced with a mesh cage. Intraoperative cystic lesions had a white crystal-like appearance (Figure-2A), with white granular tissue infiltrating the entire vertebral body and canal, thus disrupting the integrity of the vertebral body. The pathological results showed a large number of inflammatory cells and macrophage invasion in the vertebral body, consistent with the pathological damage caused by hydatid disease (Figure-2B). Before the end of the operation, the surgical site was irrigated with a hypertonic sodium chloride solution to prevent hydatid recurrence. The patient was postoperatively treated with pharmacotherapy. During the treatment period, hematopoietic and liver enzyme changes were monitored according to the treatment guidelines. The patient had a good recovery state of both lower limb powers, along with no signs of local recurrence at the latest examination one year postoperatively.\u003c/p\u003e"},{"header":"Discussion","content":"\u003cp\u003eSpinal hydatidosis is a rare disease which caused bone infection in adults. Because spinal hydatidosis has no special characteristic signs or symptoms, it is difficult to diagnose first and may be misdiagnosed as other common diseases such as lumbar intervertebral protrusion and lumbar spinal stenosis. Previous reports have shown that spinal hydatidosis occurs in 60% in thoracic vertebrae, 35% in lumbosacral vertebrae, and 5% in cervical vertebrae respectively [\u003cspan citationid=\"CR7\" class=\"CitationRef\"\u003e7\u003c/span\u003e]. The clinical symptoms of vertebral echinococcosis mainly include lumbar pain (59.2%-75%) accompanied by lower extremity weakness and paresthesia (37%-50%) [\u003cspan citationid=\"CR8\" class=\"CitationRef\"\u003e8\u003c/span\u003e, \u003cspan citationid=\"CR9\" class=\"CitationRef\"\u003e9\u003c/span\u003e]. In the present case, lower back pain and decreased power in both lower limbs accompanied paresthesia. We first misdiagnosed the patient with bone tumors or tuberculosis because of an elevated CEA level, a positive examination for tuberculosis, and vertebral body destruction. Due to the absence of signs on ECT scans and the patient history of animal herding in pastoral areas combined with the MRI images, we temporarily excluded bone tumors and considered the possibility of echinococcosis infection.\u003c/p\u003e \u003cp\u003eThe spinal cystic echinococcosis showed no characteristic signs or symptoms. Therefore, these cases are rarely detected and diagnosed in the early stages, until symptoms of vertebral destruction and compression of the spinal cord and nerve caused by enlarged cysts appear. Echinococcus diagnosis should always be considered the diagnosis of echinococcus for patients living in rural areas, especially in those in contact with animals. Previous studies have shown that spinal hydatid disease is mainly caused by echinococcus granulosus, which leads to its cystic form, resulting in spinal pathological changes [\u003cspan citationid=\"CR10\" class=\"CitationRef\"\u003e10\u003c/span\u003e]. Computed tomography (CT) and magnetic resonance imaging (MRI) are the primary imaging modalities used to diagnose spinal hydatid disease. According to the CT and MRI findings, spinal hydatid can be divided into five radiological types: intramedullary, intradural extramedullary, epidural, vertebral echinococcus, and paravertebral lesions extending into spinal structures [\u003cspan citationid=\"CR11\" class=\"CitationRef\"\u003e11\u003c/span\u003e]. Among the five radiological types, vertebral echinococcus and paravertebral lesions extending into the spinal structures are more common and are seen in the majority of patients [\u003cspan citationid=\"CR11\" class=\"CitationRef\"\u003e11\u003c/span\u003e]. Zhang et al. [\u003cspan citationid=\"CR12\" class=\"CitationRef\"\u003e12\u003c/span\u003e] found the MRI feature of spinal hydatid disease presented as a \u0026ldquo;bunch of grapes\u0026rdquo; was a typical imaging and the total vertebral resection had a better therapeutic effect than subtotal vertebral resection. The imaging examination in our patient showed that the lesions of spinal hydatid were mainly located in L2. Moreover, the MRI images of the patient we reported to show polycystic lobulated clusters of grape beads, which is consistent with previous reports [\u003cspan citationid=\"CR12\" class=\"CitationRef\"\u003e12\u003c/span\u003e].\u003c/p\u003e \u003cp\u003eThe theoretical cure for spinal hydatidosis is resection of the lesion and a preference for recurrence, but the cure rate is low when surgical intervention is performed alone. The main goal of surgical intervention for spinal echinococcosis is to completely remove the damaged part of the vertebral body and achieve healthy vertebral margins on both sides of the resection [\u003cspan citationid=\"CR13\" class=\"CitationRef\"\u003e13\u003c/span\u003e]. Zhang et al. [\u003cspan citationid=\"CR12\" class=\"CitationRef\"\u003e12\u003c/span\u003e] found the total vertebrotomy combined with achieved antiparasitic drug therapy achieved a better therapeutic effect, moreover they found antiparasitic drug play an important role in preventing recurrence. Some studies have also reported that preoperative combined with postoperative antiparasitic drugs is an important adjuvant treatment for its recurrence [\u003cspan citationid=\"CR6\" class=\"CitationRef\"\u003e6\u003c/span\u003e, \u003cspan citationid=\"CR12\" class=\"CitationRef\"\u003e12\u003c/span\u003e]. In our case, we found that subtotal vertebral resection could completely remove hydatid cysts without any residue. During surgery, we carefully and integrally removed the cyst and rinsed and soaked the wound with hypertonic saline to prevent recurrence. However, it is still unclear whether recurrence can be prevented. Subsequently, the patient was treated with antiparasitic drug therapy as an important supplementary therapy to prevent recurrence. The patient recovered well and had normal power in both lower limbs without low back pain at the last follow-up.\u003c/p\u003e"},{"header":"Conclusion","content":"\u003cp\u003eWe report a rare case of typical spinal hydatidosis treated by subtotal vertebral resection combined use of antiparasitic drugs and achieved a good therapeutic effect. Surgical removal combined with antiviral drugs is an effective treatment for spinal hydatidosis.\u003c/p\u003e"},{"header":"Abbreviations","content":"\u003cdiv class=\"DefinitionList\"\u003e \u003cdiv class=\"DefinitionListEntry\"\u003e \u003cdiv class=\"Term\"\u003eCT\u003c/div\u003e \u003cdiv class=\"Description\"\u003e \u003cp\u003ecomputed tomography\u003c/p\u003e \u003c/div\u003e \u003c/div\u003e \u003cdiv class=\"DefinitionListEntry\"\u003e \u003cdiv class=\"Term\"\u003eMRI\u003c/div\u003e \u003cdiv class=\"Description\"\u003e \u003cp\u003emagnetic resonance imaging\u003c/p\u003e \u003c/div\u003e \u003c/div\u003e \u003cdiv class=\"DefinitionListEntry\"\u003e \u003cdiv class=\"Term\"\u003eCRP\u003c/div\u003e \u003cdiv class=\"Description\"\u003e \u003cp\u003eC-reactive protein\u003c/p\u003e \u003c/div\u003e \u003c/div\u003e \u003cdiv class=\"DefinitionListEntry\"\u003e \u003cdiv class=\"Term\"\u003eCEA\u003c/div\u003e \u003cdiv class=\"Description\"\u003e \u003cp\u003ecarcinoembryonic antigen\u003c/p\u003e \u003c/div\u003e \u003c/div\u003e \u003cdiv class=\"DefinitionListEntry\"\u003e \u003cdiv class=\"Term\"\u003eL2\u003c/div\u003e \u003cdiv class=\"Description\"\u003e \u003cp\u003elumbar2\u003c/p\u003e \u003c/div\u003e \u003c/div\u003e \u003cdiv class=\"DefinitionListEntry\"\u003e \u003cdiv class=\"Term\"\u003eECT\u003c/div\u003e \u003cdiv class=\"Description\"\u003e \u003cp\u003eemission computed tomography.\u003c/p\u003e \u003c/div\u003e \u003c/div\u003e \u003c/div\u003e"},{"header":"Declarations","content":"\u003cp\u003eWe declared that all the methods and clinical data in this study was approved by the medical ethical committee at Second Hospital of Lanzhou University (2023A\u0026mdash;160). All the authors provided written informed consent to participate in this study.\u003c/p\u003e \u003cp\u003e \u003cstrong\u003eConsent for publication\u003c/strong\u003e \u003cp\u003e Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.\u003c/p\u003e \u003c/p\u003e\u003cp\u003e \u003ch2\u003eConflicts of interest\u003c/h2\u003e \u003cp\u003eThe authors declare that there are no conflicts of interest.\u003c/p\u003e \u003c/p\u003e\u003cp\u003e \u003ch2\u003eEthical approval\u003c/h2\u003e \u003cp\u003eThe medical ethical committee at Second Hospital of Lanzhou University (No. 2023A\u0026mdash;160).\u003c/p\u003e \u003c/p\u003e\u003cp\u003e \u003ch2\u003eSupplementary data\u003c/h2\u003e \u003cp\u003eNone.\u003c/p\u003e \u003c/p\u003e\u003ch2\u003eFunding\u003c/h2\u003e \u003cp\u003eThis study was supported by Key R\u0026amp;D Plan of Gansu Provincial Department of Science and Technology-Social Development Projects (Grant No. 23YFFA0043) and the Natural Science Foundation of Wuxi City (No. k20231060).\u003c/p\u003e\u003ch2\u003eAuthor Contribution\u003c/h2\u003e\u003cp\u003eRuijun Bai and Min Li conceived the project. Jinchao Cheng and Ruijun Bai drafted the manuscript. Ruijun Bai and Min Li performed the statistical analyses. Jinchao Cheng, Bing Ma, Kai Zhao, Haifeng Li revised the manuscript. All authors reviewed the manuscript. All authors read and approved the final manuscript.\u003c/p\u003e\u003ch2\u003eAcknowledgement\u003c/h2\u003e\u003cp\u003eWe thank the patient and members of the team.\u003c/p\u003e\u003ch2\u003eData Availability\u003c/h2\u003e\u003cp\u003eThe data analyzed in the study are available from the corresponding author upon reasonable request.\u003c/p\u003e"},{"header":"References","content":"\u003col\u003e\u003cli\u003e\u003cspan\u003eAnnamalai S, Muthu S, Thakur A, Ramakrishnan E. Novel Multimodal Treatment Regimen for the Management of Primary Sacrococcygeal Cystic Echinococcosis. 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World J Clin Cases. 2021;9 33:10337\u0026ndash;44; doi: \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003e10.12998/wjcc.v9.i33.10337\u003c/span\u003e\u003cspan address=\"10.12998/wjcc.v9.i33.10337\" targettype=\"DOI\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eCaglar YS, Ozgural O, Zaimoglu M, Kilinc C, Eroglu U, Dogan I, et al. Spinal Hydatid Cyst Disease: Challenging Surgery - an Institutional Experience. J Korean Neurosurg Soc. 2019;62 2:209\u0026ndash;16; doi: \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003e10.3340/jkns.2017.0245\u003c/span\u003e\u003cspan address=\"10.3340/jkns.2017.0245\" targettype=\"DOI\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e.\u003c/span\u003e\u003c/li\u003e\u003c/ol\u003e"}],"fulltextSource":"","fullText":"","funders":[],"hasAdminPriorityOnWorkflow":false,"hasManuscriptDocX":true,"hasOptedInToPreprint":true,"hasPassedJournalQc":"","hasAnyPriority":false,"hideJournal":false,"highlight":"","institution":"","isAcceptedByJournal":true,"isAuthorSuppliedPdf":false,"isDeskRejected":"","isHiddenFromSearch":false,"isInQc":false,"isInWorkflow":false,"isPdf":false,"isPdfUpToDate":true,"isWithdrawnOrRetracted":false,"journal":{"display":true,"email":"[email protected]","identity":"discover-medicine","isNatureJournal":false,"hasQc":true,"allowDirectSubmit":false,"externalIdentity":"","sideBox":"Learn more about [Discover Medicine](https://link.springer.com/journal/44337)","snPcode":"44337","submissionUrl":"https://submission.springernature.com/new-submission/44337/3","title":"Discover Medicine","twitterHandle":"","acdcEnabled":true,"dfaEnabled":true,"editorialSystem":"stoa","reportingPortfolio":"Discover Series","inReviewEnabled":true,"inReviewRevisionsEnabled":true},"keywords":"Hydatid disease, Spine, MRI imaging, Surgery, Therapy","lastPublishedDoi":"10.21203/rs.3.rs-4914019/v1","lastPublishedDoiUrl":"https://doi.org/10.21203/rs.3.rs-4914019/v1","license":{"name":"CC BY 4.0","url":"https://creativecommons.org/licenses/by/4.0/"},"manuscriptAbstract":"\u003ch2\u003eBackground\u003c/h2\u003e \u003cp\u003eHydatid disease is an infectious disease caused by echinococcus that causes liver and lung damage in humans. Bone involvement is rare and accounts for only 0.5%-4% of hydatid diseases; the spine is more prone to be infected with a proportion of 50% in musculoskeletal tissue. Missed hydatid cysts may lead to serious consequence, it is necessary to diagnosis and treatment of hydatid cysts at early stage.\u003c/p\u003e\u003ch2\u003eCase presentation:\u003c/h2\u003e \u003cp\u003eWe present a case of a 47yearold Han Chinese male diagnosed with spinal hydatidosis. The patient lived in pasturing areas accompanied by symptoms of low back pain radiating to the legs, progressive weakness, and hypoesthesia in the lower limbs accompanied by difficulty walking. Computed tomography (CT) images showed a spinal hydatid cyst in the lumbar vertebra2 (L2) that caused bone destruction in the vertebral body and lamina. Magnetic resonance imaging (MRI) images showed hyperintense cystic components on L2 with the appearance of a lobulated, multiocular, septate cystic mass and compression of the spinal cord at the segments of L2. The patient underwent subtotal vertebral resection to completely remove the damaged vertebral and paravertebral cysts, and histopathological examination revealed the characteristic features of hydatid cysts. The patient received a postoperative antiparasitic drug treatment and finally achieved a successful treatment.\u003c/p\u003e\u003ch2\u003eConclusions\u003c/h2\u003e \u003cp\u003eSpinal hydatidosis is a rare infectious disease. It should be considered as an important differential diagnosis for patients accompanying by spinal bone destruction with animal contact history. Surgical removal combined with antiviral drugs is an effective way to treat spinal hydatidosis. In addition, long term follow-up is mandatory for avoiding its recurrence.\u003c/p\u003e","manuscriptTitle":"Hydatid Disease Contributes to Spinal Cord Compression: A Case Report and Literature Review","msid":"","msnumber":"","nonDraftVersions":[{"code":1,"date":"2024-10-17 17:46:59","doi":"10.21203/rs.3.rs-4914019/v1","editorialEvents":[{"type":"communityComments","content":0},{"type":"decision","content":"Revision requested","date":"2024-10-16T06:11:41+00:00","index":"","fulltext":""},{"type":"reviewerAgreed","content":"39412273035280619816478761126689624540","date":"2024-09-30T08:46:10+00:00","index":"hide","fulltext":""},{"type":"editorInvitedReview","content":"","date":"2024-09-27T13:14:50+00:00","index":"hide","fulltext":""},{"type":"reviewerAgreed","content":"142357448074707178209423719330203794608","date":"2024-09-25T06:45:30+00:00","index":"hide","fulltext":""},{"type":"reviewerAgreed","content":"131657160025984688914808601760733442899","date":"2024-09-09T08:10:31+00:00","index":"hide","fulltext":""},{"type":"editorInvitedReview","content":"","date":"2024-09-07T15:46:35+00:00","index":"hide","fulltext":""},{"type":"reviewerAgreed","content":"338559827029451269904270176369647438680","date":"2024-09-02T13:30:51+00:00","index":"hide","fulltext":""},{"type":"reviewersInvited","content":"","date":"2024-08-28T06:55:19+00:00","index":"","fulltext":""},{"type":"editorAssigned","content":"","date":"2024-08-28T06:43:05+00:00","index":"","fulltext":""},{"type":"checksComplete","content":"","date":"2024-08-28T06:41:56+00:00","index":"","fulltext":""},{"type":"submitted","content":"Discover Medicine","date":"2024-08-14T13:31:52+00:00","index":"","fulltext":""}],"status":"published","journal":{"display":true,"email":"[email protected]","identity":"discover-medicine","isNatureJournal":false,"hasQc":true,"allowDirectSubmit":false,"externalIdentity":"","sideBox":"Learn more about [Discover Medicine](https://link.springer.com/journal/44337)","snPcode":"44337","submissionUrl":"https://submission.springernature.com/new-submission/44337/3","title":"Discover Medicine","twitterHandle":"","acdcEnabled":true,"dfaEnabled":true,"editorialSystem":"stoa","reportingPortfolio":"Discover Series","inReviewEnabled":true,"inReviewRevisionsEnabled":true}}],"origin":"","ownerIdentity":"9771d91c-c3de-43db-807a-9c18143b100e","owner":[],"postedDate":"October 17th, 2024","published":true,"recentEditorialEvents":[],"rejectedJournal":[],"revision":"","amendment":"","status":"under-review","subjectAreas":[],"tags":[],"updatedAt":"2024-10-23T07:39:22+00:00","versionOfRecord":[],"versionCreatedAt":"2024-10-17 17:46:59","video":"","vorDoi":"","vorDoiUrl":"","workflowStages":[]},"version":"v1","identity":"rs-4914019","journalConfig":"researchsquare"},"__N_SSP":true},"page":"/article/[identity]/[[...version]]","query":{"redirect":"/article/rs-4914019","identity":"rs-4914019","version":["v1"]},"buildId":"qtupq5eGEP_6zYnWcrvyt","isFallback":false,"isExperimentalCompile":false,"dynamicIds":[84888],"gssp":true,"scriptLoader":[]}