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1.  Idiopathic spinal epidural lipomatosis: urgent decompression in an atypical case 
European Spine Journal  2007;17(Suppl 2):225-227.
Symptomatic spinal epidural lipomatosis (SEL) is very rare and frequently associated to chronic exogenous steroid use, obesity and Cushing syndrome. The idiopathic cases where no identifiable association with SEL are found constitute only 17% of all cases. The usual clinical manifestations of this entity consist of dorsal or lumbar pain with paresthesias and weakness in lower limbs, but acute symptoms of myelopathy are exceptional. We report a case of acute paraparesis and urinary retention caused by thoracic SEL in a 55-year-old male who did not have any recognized predisposing factor for this condition. Urgent surgical decompression was performed in order to relieve the symptoms. Slow but progressive improvement was assessed after surgery. We consider this case to be exceptional due to the needing to perform an urgent decompressive laminectomy to treat a rapidly progressive myelopathy caused by idiopathic SEL.
doi:10.1007/s00586-007-0465-0
PMCID: PMC2525894  PMID: 17876611
Epidural lipomatosis; Paraparesis; Spinal cord compression; Surgical decompression; Myelopathy
2.  Unusual presentation of spinal lipomatosis 
Spinal epidural lipomatosis (SEL) is a rare condition characterized by overgrowth of normal adipose tissue in the extradural space within the spinal canal that can lead to significant spinal cord compression. It is most commonly reported in patients receiving chronic glucocorticoid therapy. Other causes can include obesity and hypercortisolism. Occasionally, idiopathic SEL will occur in patients with no known risk factors, but cases are more generally reported in obesity and males. We present the case of a 35 year-old non-obese woman found to have rapidly progressive SEL that was not associated with any of the common causes of the disorder.
doi:10.2147/IMCRJ.S54456
PMCID: PMC4181744  PMID: 25285024
lipomatosis; laminectomy; hypercortisolism
3.  Spinal Epidural Lipomatosis in Korean 
Spinal epidural lipomatosis (SEL) is a rare disorder, regarded in literature as a consequence of administration of exogenous steroids, associated with a variety of systemic diseases, endocrinopathies and the Cushing's syndrome. Occasionally, SEL may occur in patients not exposed to steroids or suffering from endocrinopathies, namely, idiopathic SEL. Thus far, case studies of SEL among Korean have been published rather sporadically. We reviewed the clinical features of SEL cases, among Koreans with journal review, including this report of three operated cases. According to this study, there were some differences between Korean and western cases. Koreans had higher incidences of idiopathic SEL, predominant involvement in the lumbar segments, very few thoracic involvement and lower MBI, as opposed to westerners.
doi:10.3340/jkns.2014.55.6.365
PMCID: PMC4166335  PMID: 25237435
Spinal epidural lipomatosis; Korean; Idiopathic
4.  Spinal epidural lipomatosis in children with hematologic malignancies 
Annals of hematology  2011;90(9):1067-1074.
Summary
Background
Abnormal fat deposition in the epidural space or spinal epidural lipomatosis (SEL) due to corticosteroid treatment or obesity may cause obstruction to cerebrospinal fluid flow. Little is known about SEL in patients with hematologic malignancies who require frequent lumbar punctures and corticosteroid treatment that places them at risk.
Patients and methods
Records and radiologic images of patients with SEL and leukemia or non Hodgkin lymphoma (NHL) treated at a single institution from 1999–2009 were reviewed. Risk factors were compared with 405 control patients with leukemia.
Results
Fourteen patients with leukemia or NHL were diagnosed with SEL. The majority of patients underwent diagnostic imaging after unsuccessful lumbar punctures within one month of their primary diagnosis. Prior to SEL diagnosis, all patients received systemic and/or intrathecal corticosteroids. SEL diagnosis led to modification of intrathecal administration in 8 patients, including Ommaya reservoir placement in 4 patients. All patients completed protocol-specified chemotherapy without neurologic symptoms or surgical intervention. Risk factors for developing SEL include older age and high body mass index.
Conclusions
Investigation for SEL in leukemia or lymphoma patients with difficult lumbar punctures is warranted. Placement of an Ommaya reservoir may facilitate safe CNS-directed therapy in severely affected patients.
doi:10.1007/s00277-011-1183-6
PMCID: PMC3169011  PMID: 21340722
spinal epidural lipomatosis; lumbar canal obstruction; difficult lumbar puncture; leukemia; lymphoma
5.  Steroid-induced Spinal Epidural Lipomatosis after Suprasella Tumor Resection 
Korean Journal of Spine  2013;10(2):88-90.
Spinal epidural lipomatosis (SEL) is an abnormal localized accumulation of fat tissues in the epidural space. It is strongly related with steroid administration. The symptoms of SEL are various and range from back pain to paraplegia. In severe cases, decompressive laminectomy is the choice of treatment. A 32-year-old woman who had been under long-term steroid administration after suprasellar tumor resection was admitted for both leg radiating pain and weakness. She was diagnosed with SEL and had a decompressive laminectomy. During the operation, we found the nerve roots were compressed by epidural fat tissues and engorged vessels. After the operation, her radiating pain was relieved and motor weakness was improved.
doi:10.14245/kjs.2013.10.2.88
PMCID: PMC3941716  PMID: 24757466
Spinal epidural lipomatosis; Suprasellar tumor; Steroid
6.  Rapid progression of spinal epidural lipomatosis 
European Spine Journal  2011;21(Suppl 4):408-412.
Spinal epidural lipomatosis (SEL) is a rare but well-recognized condition. In general, the onset of its symptoms is insidious and the disease progresses slowly. We report two cases of rapid progression of SEL with no history of steroid intake in non-obese individuals after epidural steroid injection. These SEL patients developed neurologic symptoms after less than 5 months; these symptoms were confirmed to be due to SEL by serial MR images. After the debulking of the epidural fat, their symptoms improved.
doi:10.1007/s00586-011-1855-x
PMCID: PMC3369035  PMID: 21667131
Spinal epidural lipomatosis; Epidural steroid injection
7.  Spinal epidural lipomatosis: An unusual cause of relapsing and remitting paraparesis 
Epidural lipomatosis is a rare entity to cause spinal cord compression and neurological deficits. This is usually associated with excess of steroids in the body either because of endogenous source as in Cushings disease or exogenous intake as in some diseases like systemic lupus erythematosus, in some endocrinopathies or in morbid obesity. But in some cases no cause has been found. Such idiopathic cases of spinal epidural lipomatosis have also been reported. Here, we report a case of idiopathic spinal epidural lipomatosis with relapsing and remitting paraparesis which is quite unusual. Treatment depends upon neurological status of the patient. We operated the patient as he had significant neurological compromise and he improved significantly.
doi:10.4103/1817-1745.76117
PMCID: PMC3087997  PMID: 21559166
Idiopathic cord compression; myelopathy; paraparesis; spinal epidural lipomatosis
8.  Solitary spinal epidural cavernous angioma: report of nine surgically treated cases and review of the literature 
European Spine Journal  2012;22(3):542-547.
Purpose
We intend to report the largest series of spinal epidural cavernomas (SEC), discuss their clinical features, imaging characteristics, surgical findings, outcome analysis and compare them with similar reports in the literature.
Methods
Among the cases of spinal tumors treated surgically by the authors, there have been nine cases of SEC’s. All the data were collected prospectively and the cases have been followed after surgery up to the time of this analysis.
Results
There were six female and three male patients with the ages ranging between 13 and 74 years. The lesions were located in the thoracic spine (4 cases), lumbar spine (4 cases) and one at the sacral level. Clinical presentations included acute spinal pain and paraparesis in two, low back pain and radiculopathy in five, and slowly progressive myelopathy in the other two cases. The lesion was iso-intense with the spinal cord in T1W images and hyperintense in T2W images and showed strong homogeneous enhancement after contrast medium injection in most of our cases. In the presence of hemorrhage inside the lesion, it was hyperintense in both T1W and T2W MR sequences as in our case 6. In the single case presenting with acute hemorrhage, epidural hematoma was the only finding, our case 1. Complete surgical removal was achieved in all our cases, and confirmed by postoperative MRI.
Conclusion
SEC is hard to be differentiated from other epidural spinal lesions before intervention but should be considered in the list of differential diagnosis regarding its favorable outcome.
doi:10.1007/s00586-012-2526-2
PMCID: PMC3585645  PMID: 23053760
Cavernous angioma; Magnetic resonance imaging; Spinal epidural cavernoma; Spinal epidural lesion
9.  Principles of management of osteometabolic disorders affecting the aging spine 
European Spine Journal  2003;12(Suppl 2):S113-S131.
Osteoporosis is the most common contributing factor of spinal fractures, which characteristically are not generally known to produce spinal cord compression symptoms. Recently, an increasing number of medical reports have implicated osteoporotic fractures as a cause of serious neurological deficit and painful disabling spinal deformities. This has been corroborated by the present authors as well. These complications are only amenable to surgical management, requiring instrumentation. Instrumenting an osteoporotic spine, although a challenging task, can be accomplished if certain guidelines for surgical techniques are respected. Neurological deficits respond equally well to an anterior or posterior decompression, provided this is coupled with multisegmental fixation of the construct. With the steady increase in the elderly population, it is anticipated that the spine surgeon will face serious complications of osteoporotic spines more frequently. With regard to surgery, however, excellent correction of deformities can be achieved, by combining anterior and posterior approaches. Paget's disease of bone (PD) is a non-hormonal osteometabolic disorder and the spine is the second most commonly affected site. About one-third of patients with spinal involvement exhibit symptoms of clinical stenosis. In only 12–24% of patients with PD of the spine is back pain attributed solely to PD, while in the majority of patients, back pain is either arthritic in nature or a combination of a pagetic process and coexisting arthritis. In this context, one must be certain before attributing low back pain to PD exclusively, and antipagetic medical treatment alone may be ineffective. Neural element dysfunction may be attributed to compressive myelopathy by pagetic bone overgrowth, pagetic intraspinal soft tissue overgrowth, ossification of epidural fat, platybasia, spontaneous bleeding, sarcomatous degeneration and vertebral fracture or subluxation. Neural dysfunction can also result from spinal ischemia when blood is diverted by the so-called "arterial steal syndrome". Because the effectiveness of pharmacologic treatment for pagetic spinal stenosis has been clearly demonstrated, surgical decompression should only be instituted after failure of antipagetic medical treatment. Surgery is indicated as a primary treatment when neural compression is secondary to pathologic fractures, dislocations, spontaneous epidural hematoma, syringomyelia, platybasia, or sarcomatous transformation. Five classes of drugs are available for the treatment of PD. Bisphosphonates are the most popular antipagetic drug and several forms have been investigated.
doi:10.1007/s00586-003-0600-5
PMCID: PMC3591829  PMID: 14505119
Osteoporosis; Fractures; Neurological deficit; Deformity; Paget's disease; Back pain; Spinal stenosis; Myelopathy; Treatment
10.  Corticosteroid-induced spinal epidural lipomatosis in the pediatric age group: report of a new case and updated analysis of the literature 
Spinal epidural lipomatosis is a rare complication of chronic corticosteroid treatment. We report a new pediatric case and an analysis of this and 19 pediatric cases identified in the international literature. The youngest of these combined 20 patients was 5 years old when lipomatosis was diagnosed. Lipomatosis manifested after a mean of 1.3 (+/- 1.5) years (SD) (median, 0.8 years; range, 3 weeks - 6.5 years) of corticosteroid treatment. The corticosteroid dose at the time of presentation of the lipomatosis ranged widely, between 5 and 80 mg of prednisone/day. Back pain was the most common presenting symptom. Imaging revealed that lipomatosis almost always involved the thoracic spine, extending into the lumbosacral region in a subset of patients. Predominantly lumbosacral involvement was documented in only two cases. Although a neurological deficit at presentation was documented in about half of the cases, surgical decompression was not performed in the cases reported after 1996. Instead, reducing the corticosteroid dose (sometimes combined with dietary restriction to mobilize fat) sufficed to induce remission. In summary, pediatric spinal epidural lipomatosis remains a potentially serious untoward effect of corticosteroid treatment, which, if recognized in a timely manner, can have a good outcome with conservative treatment.
doi:10.1186/1546-0096-9-5
PMCID: PMC3041993  PMID: 21284882
11.  Acute spinal cord compression due to epidural lipomatosis complicated by an abscess: magnetic resonance and pathology findings 
European Spine Journal  2010;19(Suppl 2):216-219.
A 68-year-old male presented with rapidly progressive paraplegia. MR images of the thoracic spine were interpreted as being consistent with an abscess within an epidural lipomatosis compressing the spinal cord. Laminectomy was performed, and a large amount of pus was drained from the epidural lipomatosis, from which Staphylococcus aureus was isolated. This is the first reported case of an abscess involving an epidural lipomatosis.
doi:10.1007/s00586-010-1393-y
PMCID: PMC2899645  PMID: 20372939
Epidural lipomatosis; Spinal abscess; Magnetic resonance imaging
12.  Idiopathic Hypertrophic Spinal Pachymeningitis : Report of Two Cases and Review of the Literature 
Idiopathic hypertrophic spinal pachymeningitis (IHSP) is a rare inflammatory disease characterized by hypertrophic inflammation of the dura mater and various clinical courses that are from myelopathy. Although many associated diseases have been suggested, the etiology of IHSP is not well understood. The ideal treatment is controversial. In the first case, a 55-year-old woman presented back pain, progressive paraparesis, both leg numbness, and voiding difficulty. Initial magnetic resonance imaging (MRI) demonstrated an anterior epidural mass lesion involving from C6 to mid-thoracic spine area with low signal intensity on T1 and T2 weighted images. We performed decompressive laminectomy and lesional biopsy. After operation, she was subsequently treated with steroid and could walk unaided. In the second case, a 45-year-old woman presented with fever and quadriplegia after a spine fusion operation due to lumbar spinal stenosis and degenerative herniated lumbar disc. Initial MRI showed anterior and posterior epidural mass lesion from foramen magnum to C4 level. She underwent decompressive laminectomy and durotomy followed by steroid therapy. However, her conditions deteriorated gradually and medical complications occurred. In our cases, etiology was not found despite through investigations. Initial MRI showed dural thickening with mixed signal intensity on T1- and T2-weighted images. Pathologic examination revealed chronic nonspecific inflammation in both patients. Although one patient developed several complications, the other showed slow improvement of neurological symptoms with decompressive surgery and steroid therapy. In case of chronic compressive myelopathy due to the dural hypertrophic change, decompressive surgery such as laminectomy or laminoplasty may be helpful as well as postoperative steroid therapy.
doi:10.3340/jkns.2011.50.4.392
PMCID: PMC3243847  PMID: 22200026
Idiopathic hypertrophic spinal pachymeningitis; Spinal cord compression; Chronic nonspecific inflammation; Dural thickening
13.  Chordoma of the thoracic spine in an 89-year-old 
European Spine Journal  2011;21(Suppl 4):428-432.
Study design
Case report and literature review.
Objective
Report the highly unusual presentation of thoracic spine chordoma in an 89-year-old and review existing literature as it impacts treatment in the elderly.
Summary of background data
Chordomas are infrequent tumors of the spine that commonly present during mid-adulthood at the spheno-occipital or sacrococcygeal junctions. The mobile spine is affected in 10–15% of cases but chordomas are extremely rare in the thoracic spine. Chordoma rarely enters the differential diagnosis of spinal tumors in elderly patients, for whom metastases and multiple myeloma are by far the most common.
Methods
A case report is detailed of an 89-year-old male presenting with incapacitating pain and early signs of thoracic myelopathy. A lytic, expanding lesion of the T10 vertebral body with epidural spinal cord compression was identified. In the absence of evidence of other primary tumor, a CT-guided needle biopsy revealed chordoma. A literature review of reported thoracic spine chordomas was also performed.
Results
An intralesional posterolateral resection and reconstruction was performed with good results and no recurrence at 13 months’ follow-up. 30 reports of thoracic spine chordomas were identified in the literature since 1902. Mean age of presentation at 35.7 years is earlier than for most chordomas. The oldest previously reported patient was 68-year-old. Neurological impairment at presentation is rare with the usual presenting symptom being pain. Response to radiation and chemotherapy is limited. 5-year survival rates range from 50 to 60%.
Conclusion
Chordoma is a highly uncommon epidural neoplasm of the thoracic spine and is vanishingly rare in elderly patients. This report documents the oldest reported patient with thoracic spine chordoma at 89-year-old. Their locally aggressive behavior typically prompts consideration of aggressive surgical resection. When tailored to the individual patient, such procedures are feasible without excessive morbidity even in elderly patients.
doi:10.1007/s00586-011-1980-6
PMCID: PMC3369041  PMID: 21866405
Chordoma; Spine; Vertebrae, thoracic; Corpectomy
14.  Spinal Cord Stimulation for Neuropathic Pain 
Executive Summary
Objective
The objective of this health technology policy assessment was to determine the effectiveness of spinal cord stimulation (SCS) to manage chronic intractable neuropathic pain and to evaluate the adverse events and Ontario-specific economic profile of this technology.
Clinical Need
SCS is a reversible pain therapy that uses low-voltage electrical pulses to manage chronic, intractable neuropathic pain of the trunk or limbs. Neuropathic pain begins or is caused by damage or dysfunction to the nervous system and can be difficult to manage.
The prevalence of neuropathic pain has been estimated at about 1.5% of the population in the United States and 1% of the population in the United Kingdom. These prevalence rates are generalizable to Canada.
Neuropathic pain is extremely difficult to manage. People with symptoms that persist for at least 6 months or who have symptoms that last longer than expected for tissue healing or resolution of an underlying disease are considered to have chronic pain. Chronic pain is an emotional, social, and economic burden for those living with it. Depression, reduced quality of life (QOL), absenteeism from work, and a lower household income are positively correlated with chronic pain.
Although the actual number is unknown, a proportion of people with chronic neuropathic pain fail to obtain pain relief from pharmacological therapies despite adequate and reasonable efforts to use them. These people are said to have intractable neuropathic pain, and they are the target population for SCS.
The most common indication for SCS in North America is chronic intractable neuropathic pain due to failed back surgery syndrome (FBSS), a term that describes persistent leg or back and leg pain in patients who have had back or spine surgery. Neuropathic pain due to complex regional pain syndrome (CRPS), which can develop in the distal aspect of a limb a minor injury, is another common indication. To a lesser extent, chronic intractable pain of postherpetic neuralgia, which is a persistent burning pain and hyperesthesia along the distribution of a cutaneous nerve after an attack of herpes zoster, is also managed with SCS.
For each condition, SCS is considered as a pain management therapy only after conventional pain therapies, including pharmacological, nonpharmacological, and surgical treatments, if applicable, have been attempted and have failed.
The Technology
The SCS technology consists of 3 implantable components: a pulse generator, an extension cable, and a lead (a small wire). The pulse generator is the power source for the spinal cord stimulator. It generates low-voltage electrical pulses. The extension cable connects the pulse generator to the lead. The lead is a small, insulated wire that has a set of electrodes at one end. The lead is placed into the epidural space on the posterior aspect of the spinal cord, and the electrodes are positioned at the level of the nerve roots innervating the painful area. An electrical current from the electrodes induces a paresthesia, or a tingling sensation that masks the pain.
Before SCS is initiated, candidates must have psychological testing to rule out major psychological illness, drug habituation, and issues of secondary gain that can negatively influence the success of the therapy. Successful candidates will have a SCS test stimulation period (trial period) to assess their responsiveness to SCS. The test stimulation takes about 1 week to complete, and candidates who obtain at least 50% pain relief during this period are deemed suitable to receive a permanent implantation of a spinal cord stimulator
Review Strategy
The Medical Advisory Secretariat (MAS) reviewed all published health technology assessments of spinal cord stimulation. Following this, a literature search was conducted from 2000 to January, 2005 and a systematic review of the literature was completed. The primary outcome for the systematic review was pain relief. Secondary outcomes included functional status and quality of life. After applying the predetermined inclusion and exclusion criteria, 2 randomized controlled trials (MAS level 2 evidence), and 2 prospective non-randomized controlled trials with a before-and-after-treatment study design (MAS level 3a evidence) were retrieved and reviewed.
Summary of Findings
The authors of 6 health technology assessments concluded that evidence exists to support the effectiveness of SCS to decrease pain in various neuropathic pain syndromes. However, the quality of this evidence varied among reports from weak to moderate.
The systematic review completed by MAS found high quality level 2 evidence that SCS decreases pain and level 3a evidence that it improves functional status and quality of life in some people with neuropathic pain conditions. The rate of technical failures was approximately 11%, which included electrode lead migration and/or malposition. Procedural complications included infection and dural puncture; each occurred at a rate of 1.2%.
Conclusions
SCS may be considered for patients with chronic, neuropathic pain for whom standard pain treatments have failed and when there is no indication for surgical intervention to treat the underlying condition.
PMCID: PMC3382299  PMID: 23074473
15.  An Unusual Cause of Paraparesis in a Patient on Chronic Steroid Therapy 
Background/Objective:
Spinal epidural lipomatosis is the excessive deposition of unencapsulated fat in the epidural space. This is a rare disorder often associated with high levels of endogenous steroids or the administration of exogenous steroids.
Case Description:
A 32-year-old man with congenital kyphosis treated with prednisolone daily for 5 months for interstitial lung disease developed compressive myelopathy.
Findings:
Magnetic resonance imaging showed congenital kyphosis along with epidural lipomatosis compressing the cord. Cessation of steroid therapy was associated with improvement in the symptoms.
Conclusions:
Spinal epidural lipomatosis is a rare side effect of chronic steroid therapy that may occur with relatively short-term, low-dose regimens. In patients with congenital vertebral anomalies, spinal fat deposition may worsen the neurological status in an already compromised cord. Discontinuation of steroid therapy is beneficial; some patients may require surgical intervention for decompression.
PMCID: PMC2032003  PMID: 17385272
Lipomatosis, spinal epidural; Steroid therapy complications; Kyphosis, congenital; Prednisolone; Myelopathy, compressive; Paraparesis; Cushing syndrome
16.  The flow patterns of caudal epidural in upper lumbar spinal pathology 
European Spine Journal  2010;20(5):804-807.
Epidural steroid injections are an important therapeutic modality employed by spinal surgeons in the treatment of patients with chronic low back pain with or without lumbar radiculopathy. The caudal epidural is a commonly used and well-established technique; however, little is known about the segmental level of pathology that may be addressed by this intervention. This prospective study of over 50 patients aimed to examine the spreading pattern of this technique using epidurography. The effect of variation in Trendelenburg tilt and the eradication of lumbar lordosis on the cephalic distribution of the injectate were investigated. 52 patients with low back pain and radiculopathy underwent caudal epidural. All had 20 ml volume injected, comprised of 5 ml contrast (Ultravist™ Schering) 2 ml Triamcinolone (Adcortyl™ Squibb) and 13 ml local anaesthetic (1% lignocaine). Patients were randomised to either 0° or 30° of Trendelenburg tilt, as referenced from the lumbar spine. Patients were further randomised to presence or absence of lumbar lordosis, which was eradicated using a flexion device placed beneath the prone patient. A lateral image of each sacrum was obtained, to identify variations in sacral geometry particularly resistant to cephalic spread of injectate. The highest segment reached on fluoroscopy was recorded post injection. Fifty-two patients with a mean age of 50 years underwent caudal epidural. Thirty-one were in 0° head tilt, with 21 in 30° of head tilt. In each of these groups, 50% had their lumbar lordosis flattened prior to caudal injection. The median segmental level reached was L3, with a range from T9 to L5. Eradication of lumbar lordosis did not significantly alter cephalic spread of injectate. There was a trend for 30° tilt to extend the upper level reached by caudal injection (p = 0.08). There were no adverse events in this series. Caudal epidural is a reliable and relatively safe procedure for the treatment of low back pain. Pathology at L3/4 and L4/5 and L5/S1 can be approached by this technique. Although in selected cases thoracic and high lumbar levels can be reached, this is variable. If pathology at levels above L3 needs to be addressed, we propose a 30° head tilt may improve cephalic drug delivery. The caudal route is best reserved for pathology below L3.
doi:10.1007/s00586-010-1613-5
PMCID: PMC3082677  PMID: 21113636
Caudal epidural; Lumbar spine
17.  Dorsal epidural spinal lipomatosis 
BMJ Case Reports  2011;2011:bcr0920103365.
The authors report a case of a thoracic epidural spinal lipomatosis causing severe neurological deficits along the review of pertinent literature. The patient is a 56-year-old woman who presented with acute onset of severe paraparesis; she was investigated with cervical and thoracic MRI and then surgically managed because of an intraspinal mass compressing the cord. The operation consisted in the excision of the mass confirmed to be a fibrolipoma by pathological analysis. The patient attained complete neurological recovery and at 18 months follow-up she reported a generalised well-being. Thoracic lipomas are rare lesions that presenting mostly with back pain; however, in rare instances they may cause progressive and/or abrupt neurological dysfunction. Appropriate imaging can help in the diagnosis and management of such cases.
doi:10.1136/bcr.09.2010.3365
PMCID: PMC3062862  PMID: 22707370
18.  Spontaneous and idiopathic chronic spinal epidural hematoma: two case reports and review of the literature 
European Spine Journal  2009;18(11):1555-1561.
Spinal epidural hematoma (SEH) represents the most frequent entity of acute or chronic spinal bleeding. Based upon pathogenesis, SEH can be classified as idiopathic, spontaneous, and secondary. The idiopathic forms are considered not to be attributed to any specific risk factors. Spontaneous SEH, accounting for 0.3–0.9% of all spinal epidural space occupying lesions, instead is associated with risk factors (such as substantial soft trauma or coagulation abnormalities). The chronic form, as our literature review revealed, is the rarest and its most frequent location is the lumbar spine. The pathophysiology of spontaneous and idiopathic SEH is still under debate: There are only a few reports in literature of chronically evolving SEH with progressively increasing pain and neurological impairment. Magnetic resonance imaging may be inconclusive for differential diagnosis. Here, we present two cases of lumbar chronic SEH with slow, progressive, and persistent lumbar radicular impairment. The first patient reported a minor trauma with slight back contusion and thus was classified as spontaneous SEH. In the second case not even a minor trauma was involved, so we considered it to be idiopathic SEH. In both cases preoperative blood and coagulation tests were normal and we did not find any other or co-factors in the patients’ clinical histories. MR imaging showed uncertain spinal canal obstructing lesions at L3 and L4 level in both cases. Surgical treatment allowed a correct diagnosis and resulted in full clinical and neuroradiological recovery after 1 year follow-up. Our aim is to discuss pathogenesis, clinical and radiological features, differential diagnosis and treatment options, on the background of relevant literature review.
doi:10.1007/s00586-009-1175-6
PMCID: PMC2899401  PMID: 19859747
Spontaneous; Idiopathic; Chronic; Hematoma; Epidural; Spinal
19.  Complete motor recovery after acute paraparesis caused by spontaneous spinal epidural hematoma: case report 
Background
Spontaneous spinal epidural hematoma is a relatively rare but potentially disabling disease. Prompt timely surgical management may promote recovery even in severe cases.
Case presentation
We report a 34-year-old man with a 2-hour history of sudden severe back pain, followed by weakness and numbness over the bilateral lower limbs, progressing to intense paraparesis and anesthesia. A spinal magnetic resonance imaging scan was performed and revealed an anterior epidural hematoma of the thoracic spine. He underwent an emergency decompression laminectomy of the thoracic spine and hematoma evacuation. Just after surgery, his lower extremity movements improved. After 1 week, there was no residual weakness and ambulation without assistance was resumed, with residual paresthesia on the plantar face of both feet. After 5 months, no residual symptoms persisted.
Conclusions
The diagnosis of spontaneous spinal epidural hematoma must be kept in mind in cases of sudden back pain with symptoms of spinal cord compression. Early recognition, accurate diagnosis and prompt surgical treatment may result in significant improvement even in severe cases.
doi:10.1186/1471-227X-11-10
PMCID: PMC3160384  PMID: 21794133
20.  An intradural cervical chordoma mimicking schwannoma 
Journal of Injury and Violence Research  2012;4(3 Suppl 1): Paper No. 83.
Abstract:
Chordoma is a relatively rare tumor originating from the embryonic remnants of the notochord. This is an aggressive, slow growing and invasive tumor. It occurs mostly at the two ends of neuroaxis which is more frequent in the sacrococcygeal region. Chordoma in vertebral column is very rare. This tumor is extradural in origin and compresses neural tissues and makes the patient symptomatic. This tumor found extremely rare in the spinal region as an intradural tumor.
The present study reports a rare case of intradural chordoma tumor as well as its clinical manifestations and treatment options.
Case:
The patient was a 50-year-old female presented with 9 months history of progressively worsening neck pain, cervical spine chordoma resembling neurinoma and right arm numbness. Physical examination showed no weakness in her limbs, but she had upward plantar reflex and mild hyperreflexia. In a magnetic resonance imaging (MRI) scan of the cervical spine there was an ill-defined enhancing mass in the posterior aspect of C2-C3 body caused cord compression more severe in right side as well as foraminal scalloping. The patient underwent surgery and after midline posterior cervical incision and paravertebral muscle stripping a laminectomy was performed from C1 through C4 using a high speed drill. Needle biopsy revealed chordoma on frozen section and all of accessible parts of tumor were excised. The gross and microscopic histopathological appearance was consistent with chordoma.
Chordomas are malignant tumors that arise from remains of embryonic notochord. These ectopic rests of notochord termed “ecchordosis physaliphora “can be found in approximately 2% of autopsies. These are aggressive, slow growing, locally invasive and destructive tumors those occur in the midline of neuroaxis. They generally thought to account for 2% to 4% of all primary bone neoplasms and 1% to 4% malignant bone neoplasms. They are the most frequent primary malignant spinal tumors after plasmacytomas. The incidence has been estimated to be 0.51 cases per million. The most common location is sacrococcygeal region followed by the clivus. These two locations account for approximately 90% of chordomas. Of the tumors that do not arise in the sacrum or clivus, half occur in the cervical region, with the remainder found in the lumbar or thoracic region, in descending order of frequency. Cervical spine chordomas account for 6% of all cases. Distal metastasis most often occurs in young patients, those with sacrococcygeal or vertebral tumors, and those with atypical histological features. These tumors usually spread to contiguous anatomical structures, but they may be found in distant sites (skin, musculoskeletal system, brain, and other internal organs). Seeding of the tumor has also been reported, and the likely mechanism seems to be tumor cell of contamination during the surgical procedures. The usual radiological findings in chordomas of spine are destructive or lytic lesions with occasional sclerotic changes. They tend to lie anterolateral, rather than dorsal towards the cord, and reportedly known to invade the dura. The midline location, destructive nature, soft tissue mass formation and calcification are the radiological hallmarks of chordomas. Computed Tomography (CT) scan is the best imaging modality to delineate areas of osteolytic, osteosclerotic, or mixed areas of bone destruction.Chordoma is usually known as a hypovascular tumor which grows in a lobulated manner. Septal enhancement which reflects a lobulated growth pattern is seen in both CT and MRI and even in gross examination. Other epidural tumors include neurinoma, neurofibroma, meningioma, neuroblastoma, hemangioma, lymphoma and metastases. Their differentiation from chordoma may be difficult due to the same enhancement pattern on CT and MRI.
A dumbbell-shaped chordoma is a rare pathogenic condition. The dumbbell shape is a characteristic finding of neurinomas in spine but in spinal neurinomas extention to transverse foramina has not yet been reported. Although our case mimicked a dumbbell shaped neurogenic tumor, its midline location and destructive pattern were characteristic feature indicating a clue to the diagnosis of chordoma that was already confirmed with histopathology.
This unusual behavior of tumor extension can be explained by the soft and gelatinous nature of the tumor enabling the mass to extend or creep into the existing adjacent anatomical structures.
Keywords:
Cervical Chordoma, Intradural, Computed tomography
PMCID: PMC3571609
21.  Chronic pure radiculopathy in patient with organizing epidural hematoma around C8 nerve root 
European Spine Journal  2012;21(Suppl 4):450-452.
Spontaneously occurring spinal epidural hematomas are uncommon clinical findings, and the chronic form is the rarest and its most frequent location is the lumbar spine. Pure radicular involvement is far less frequent than myelopathy. We report a case of progressive radiculopathy in a 52-year-old man with spontaneously occurring cervical epidural hematoma (SCEH). The patient had left hand weakness and numbness for 4 months. MRI scan showed small space-occupying lesion around left 8th cervical nerve root. After surgery we confirmed chronic organizing epidural hematoma. To the best of our knowledge, this is the second reported case in the worldwide literature of pure radiculopathy in a patient with chronic SCEH.
doi:10.1007/s00586-011-2071-4
PMCID: PMC3369032  PMID: 22249307
Spontaneous epidural hematoma; Radiculopathy; Chronic
22.  Spinal cord stimulation with implanted epidural paddle lead relieves chronic axial low back pain 
Journal of Pain Research  2014;7:465-470.
Introduction
Spinal cord stimulation (SCS) provides significant relief for lumbosacral radiculopathy refractory to both medical and surgical treatment, but historically only offers limited relief for axial low back pain (LBP). We aim to evaluate the response of chronic axial LBP treated with SCS using a surgically implanted epidural paddle lead.
Materials and methods
This is a retrospective review of a consecutive series of patients with exclusive LBP or predominant LBP associated with lower extremity (LE) pain evaluated and treated with SCS using an implanted paddle lead within the dorsal thoracic epidural space. Baseline LBP, and if present LE pain, were recorded using the visual analogue scale (VAS) at an initial evaluation. At a follow-up visit (a minimum of 12 months later), LBP and LE pain after a spinal cord stimulator implantation were again recorded using the VAS. Patients were also asked to estimate total LBP pain relief achieved.
Results
Patients with either exclusive (n=7) or predominant (n=2) axial LBP were treated with SCS by implantation of a paddle lead at an average spine level of T9. The baseline VAS score for LBP was 7.2; after a follow-up of 20 months, the score decreased to 2.3 (P=0.003). The LE pain VAS score decreased from 7.5 to 0.0 (P=0.103). Patients also reported a subjective 66.4% decrease of their LBP at follow-up. There were no surgical complications.
Conclusions
Axial LBP is refractory to many treatments, including SCS. SCS using a surgically implanted paddle electrode provides significant pain relief for chronic axial LPB, and is a safe treatment modality.
doi:10.2147/JPR.S66414
PMCID: PMC4136982  PMID: 25143753
SCS; LBP; laminotomy; paddle electrodes
23.  Lumbar vertebral haemangioma causing pathological fracture, epidural haemorrhage, and cord compression: a case report and review of literature 
Context
Vertebral haemangiomas are recognized to be one of the commonest benign tumours of the vertebral column, occurring mostly in the thoracic spine. The vast majority of these are asymptomatic. Infrequently, these can turn symptomatic and cause neurological deficit (cord compression) through any of four reported mechanisms: (1) epidural extension; (2) expansion of the involved vertebra(e) causing spinal canal stenosis; (3) spontaneous epidural haemorrhage; (4) pathological burst fracture. Thoracic haemangiomas have been reported to be more likely to produce cord compression than lumbar haemangiomas.
Findings
A forty-nine year old male with acute onset spinal cord compression from a pathological fracture in a first lumbar vertebral haemangioma. An MRI delineated the haemangioma and extent of bleeding that caused the cord compression. These were confirmed during surgery and the haematoma was evacuated. The spine was instrumented from T12 to L2, and a cement vertebroplasty was performed intra-operatively. Written consent for publication was obtained from the patient.
Clinical Relevance
The junctional location of the first lumbar vertebra, and the structural weakness from normal bone being replaced by the haemangioma, probably caused it to fracture under axial loading. This pathological fracture caused bleeding from the vascularized bone, resulting in cord compression.
doi:10.1179/2045772311Y.0000000004
PMCID: PMC3127359  PMID: 21756575
Vertebral haemangioma; Pathological fracture; Spinal cord compression; Vertebroplasty; Paralysis; Paraparesis
24.  Expression of the selenoprotein S (SELS) gene in subcutaneous adipose tissue and SELS genotype are associated with metabolic risk factors 
Metabolism  2011;60(1-6):114-120.
The selenoprotein S (SELS) is a putative receptor for serum amyloid A, and recent studies have suggested that SELS may be a link between type 2 diabetes mellitus and inflammation. Genetic studies of SELS polymorphisms have revealed associations with circulating levels of inflammatory markers and hard end points of cardiovascular disease. In this study, we analyzed SELS expression in subcutaneous adipose tissue and SELS genotype in relation to metabolic risk factors. DNA microarray expression analysis was used to study the expression of SELS in lean and obese siblings from the Swedish Obese Subjects Sib Pair Study. TaqMan genotyping was used to analyze 3 polymorphisms, previously found to be associated with circulating levels of inflammatory markers, in the INTERGENE case-control study of myocardial infarction and unstable angina pectoris. Possible associations between SELS genotype and/or expression with anthropometry and measures of metabolic status were investigated. Real-time polymerase chain reaction was used to analyze the SELS expression in isolated human adipocytes incubated with insulin. In lean subjects, we found correlations between SELS gene expression in subcutaneous adipose tissue and measures of obesity (waist, P = .045; sagittal diameter, P = .031) and blood pressure (diastolic, P = .016; systolic P = .015); and in obese subjects, we found correlations with measures of obesity (body mass index, P = .03; sagittal diameter, P = .008) and glycemic control (homeostasis model assessment of insulin resistance, P = .011; insulin, P = .009) after adjusting for age and sex. The 5227GG genotype was associated with serum levels of insulin (P = .006) and homeostasis model assessment of insulin resistance (P = .007). The expression of SELS increased after insulin stimulation in isolated human adipocytes (P = .008). In this study, we found an association between both SELS gene expression in adipose tissue and SELS genotype with measures of glycemic control. In vitro studies demonstrated that the SELS gene is regulated by insulin in human subcutaneous adipocytes. This study further supports a role for SELS in the development of metabolic disease, especially in the context of insulin resistance.
doi:10.1016/j.metabol.2010.05.011
PMCID: PMC3004038  PMID: 20619427
25.  Is epidural lipomatosis associated with abnormality of body fat distribution? A case report 
European Spine Journal  2005;15(1):105-108.
Case report: To report a case of epidural lipomatosis in a patient with abnormal adipose tissue distribution, glucose intolerance and mixed hyperlipidemia. A 63-year-old male patient presented with low back pain radiating to the left calf on standing and walking (walking distance <100 m). He weighed 97.5 kg, was 1.73 m tall (BMI 32.6 kg/m2) and had a waist circumference of 113 cm. He had a glucose intolerance after a 75-g glucose oral load test. CT-Myelography revealed voluminous epidural lipomatosis around L4–L5 and L5–S1. Low calorie diet and reduction in alcohol intake achieved a weight loss of 17.5 kg in 7 months (80 kg, BMI 25.8 kg/m2, waist circumference 94 cm) and dramatic improvement in low back pain, walking distance (>500 m) and reduction of lipomatosis on CT-scan. Our case suggests a relationship between central obesity phenotype and epidural lipomatosis. Specific insulin resistance treatment might be proposed for these patients if this hypothesis is confirmed in further studies.
doi:10.1007/s00586-005-0955-x
PMCID: PMC3454559  PMID: 16007467
Lipomatosis; Epidural; Obesity; Hyperinsulinism

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