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1.  Pregnancy related symptomatic vertebral hemangioma 
Vertebral hemangiomas are benign vascular tumors of the spine that remain asymptomatic in most cases and incidentally encountered on imaging. Rarely, altered hemodynamic and hormonal changes during pregnancy may expand these benign lesions resulting in severe cord compression. The management of symptomatic vertebral hemangioma during pregnancy is controversial as modalities like radiotherapy and embolization are not suitable and surgery during pregnancy has a risk of preterm labor. Few cases of pregnancy related symptomatic vertebral hemangioma with marked epidural component have been reported in the literature. We report a case of 23-year-old primigravida who developed rapidly progressive paraparesis at 28 weeks of gestation and spine magnetic resonance imaging (MRI) revealed upper thoracic vertebral hemangioma with extensive extra-osseous extension and spinal cord compression. Laminectomy and surgical decompression of the cord was performed at 32 weeks of the pregnancy. There was significant improvement in muscle power after a week of surgery. Six weeks postoperatively she delivered a full term normal baby with subsequent improvement of neurologic deficit. Repeat MRI of dorsal spine performed at 3 months postoperatively showed reduced posterior and anterior epidural components of vertebral hemangioma.
doi:10.4103/0972-2327.128577
PMCID: PMC3992751  PMID: 24753678
Epidural extension; laminectomy; pregnancy; spinal cord compression; vertebral hemangioma
2.  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
3.  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
4.  Testicular seminomatous mixed germ cell tumor with choriocarcinoma and teratoma with secondary somatic malignancy: a case report 
Introduction
Testicular tumors are a heterogeneous group of neoplasms exhibiting diverse histopathology and can be classified as seminomatous and non-seminomatous germ cell tumor types. Mixed germ cell tumors contain more than one germ cell component and various combinations have been reported. Here, we present a rare case of a mixed germ cell tumor composed of seminoma, choriocarcinoma and teratoma with a secondary somatic malignancy.
Case presentation
A 31-year-old Caucasian man presented with splenic rupture to our hospital. A right-sided testicular swelling had been present for 6 months and his alpha-fetoprotein, beta-human chorionic gonadotropin, and lactose dehydrogenase were increased. An ultrasound of his scrotum revealed an enlarged right testis with heterogeneous echogenicity. Multiple hypervascular lesions were noted in his liver and spleen. He underwent transcatheter embolization therapy of his splenic artery followed by splenectomy and right-sided orchiectomy. A computed tomography scan also showed metastasis to both lungs. During his last follow up after four cycles of cisplatin-based chemotherapy, the level of tumor markers had decreased, decreases in the size of his liver and pulmonary lesions were noted but new sclerotic lesions were evident in his thoracolumbar region raising concern for bony metastasis.
Conclusions
Prognosis of testicular tumor depends mainly on the clinical stage, but emergence of a sarcomatous component presents a challenge in the treatment of germ cell tumors and the histological subtype of this component can be used as a guide to specific chemotherapy in these patients.
doi:10.1186/1752-1947-8-1
PMCID: PMC3917416  PMID: 24380446
Mixed germ cell tumors; Rhabdomyosarcoma; Sarcomatous component
5.  Multilevel vertebral hemangiomas: two episodes of spinal cord compression at separate levels 10 years apart 
European Spine Journal  2005;14(7):706-710.
This case report presents a 66-year-old woman with multiple vertebral hemangiomas causing spinal cord compression at different levels with a long symptom-free interval between episodes of compression. She presented with back pain and progressive weakness and numbness in her lower limbs for 3 months. Ten years earlier, she had had a symptomatic T4 vertebral hemangioma operated successfully, and had made a full recovery. Magnetic resonance imaging (MRI) of the thoracic and lumbar spine revealed multiple thoracic and lumbar vertebral hemangiomas. Extraosseous extension of a hemangioma at T9 was causing spinal cord compression. Selective embolization was performed preoperatively, and cord decompression was achieved via anterior T9 corpectomy. The patient’s neurological status improved rapidly after surgery. After a course of radiotherapy, she was neurologically intact and could walk independently. One year later, MRI showed complete resolution of the cord edema at T9, and showed regression of the high signal intensity that had been observed at unoperated levels. These findings indicated diminished vascularity and reduced aggression of the tumor.
doi:10.1007/s00586-005-0885-7
PMCID: PMC3489226  PMID: 15856339
Vertebral hemangioma; Cord compression; Embolization; Corpectomy
6.  Aneurysmal bone cysts of the spine 
European Spine Journal  2012;22(3):593-601.
Purpose
Aneurysmal bone cyst is a benign, relatively uncommon lesion, representing 1.4 % of primary bone tumors. The vertebral column is involved in 3–30 % of cases. This report describes clinical characteristics and treatment results of 18 patients with aneurysmal bone cyst of the spine.
Methods
Between 1991 and 2008, 18 patients with aneurysmal bone cyst of the spine were surgically treated in our department. The clinical records, radiographs, histologic sections, and operative reports were analyzed.
Results
There were 11 male and 7 female patients; mean age was 22.1 years (range 7–46 years). Localizations were cervical (3), cervicothoracic (2), thoracic (3), lumbar (4), and sacrum (6). Tumor was localized on the left side in 11 cases, on the right side in 2 and at midline in 5 patients. The two most common clinical features were axial pain (14 patients) and radicular pain (8 patients). Neurological signs were paraparesis in 3, monoparesis in 6. Mean duration of symptoms was 9 months (range 3 months–3 years). All patients underwent surgery: total removal was performed in 13 patients and subtotal resection in 5. Posterior (11), anterolateral (1), or combined anterior-posterior (6) approaches were used. Mean follow-up duration was 112.3 months (range 4–21 years). We detected four recurrences in subtotal excision group (4/5), and one recurrence in total excision group (1/13).
Conclusion
Treatment options for aneurysmal bone cysts are simple curettage with or without bone grafting, complete excision, embolization, radiation therapy, or a combination of these modalities. Radical surgical excision should be the goal of surgery to decrease the recurrence rate. Recurrence rate is significantly lower in case of total excision.
doi:10.1007/s00586-012-2510-x
PMCID: PMC3585636  PMID: 23053752
Aneurysmal bone cyst; Spine tumor; Spinal fusion; Tumor recurrence
7.  Posterior Reversible Encephalopathy Syndrome in a Case of Postoperative Spinal Extradural Haematoma: Case Report and Review of Literature 
Asian Spine Journal  2011;5(1):64-67.
A 14-year-old girl presented with progressive paraparesis and paresthesia of one-year duration. Magnetic resonance imaging revealed a T6 vertebral hemangioma with epidural compression on the spinal cord. Following angiography and embolization, she underwent dorsal laminectomy and excision of the soft tissue component compressing the cord. In the postoperative period she had rapid worsening of lower limb power and imaging demonstrated an epidural haematoma at the operative site. The patient was taken up for urgent re-exploration and evacuation of haematoma. Postoperatively the patient complained of visual failure, headache and had multiple episodes of seizures. An magnetic resonance imaging brain showed characteristic features of posterior reversible encephalopathy syndrome (PRES) and the patient improved gradually after control of hypertension. This is the first documented case of PRES following spinal cord compression in a patient without any known risk factors. We postulate the possible mechanism involved in its pathogenesis.
doi:10.4184/asj.2011.5.1.64
PMCID: PMC3047900  PMID: 21386948
Posterior reversible encephalopathy syndrome; Magnetic resonance imaging; Hematoma; epidural; spinal
8.  Preoperative Embolization of Hypervascular Thoracic, Lumbar, and Sacral Spinal Column Tumors: Technique and Outcomes from a Single Center 
Interventional Neuroradiology  2013;19(3):377-385.
Summary
The existing literature on preoperative spine tumor embolization is limited in size of patient cohorts and diversity of tumor histologies. This report presents our experience with preoperative embolization of hypervascular thoracic, lumbar, and sacral spinal column tumors in the largest series to date.
We conducted a retrospective review of 228 angiograms and 188 pre-operative embolizations for tumors involving thoracic, lumbar and sacral spinal column. Tumor vascularity was evaluated with conventional spinal angiography and was graded from 0 (same as normal adjacent vertebral body) to 3 (severe tumor blush with arteriovenous shunting). Embolic materials included poly vinyl alcohol (PVA) particles and detachable platinum coils and rarely, liquid embolics. The degree of embolization was graded as complete, near-complete, or partial. Anesthesia records were reviewed to document blood loss during surgery.
Renal cell carcinoma (44.2%), thyroid carcinoma (9.2%), and leiomyosarcoma (6.6%) were the most common tumors out of a total of 40 tumor histologies. Hemangiopericytoma had the highest mean vascularity (2.6) of all tumor types with at least five representative cases followed by renal cell carcinoma (2.0) and thyroid carcinoma (2.0). PVA particles were used in 100% of cases. Detachable platinum coils were used in 51.6% of cases. Complete, near-complete, and partial embolizations were achieved in 86.1%, 12.7%, and 1.2% of all cases, respectively. There were no new post-procedure neurologic deficits or other complications with long-term morbidity. The mean intra-operative blood loss for the hypervascular tumors treated with pre-operative embolization was 1745 cc.
Preoperative embolization of hypervascular thoracic, lumbar, and sacral spine tumors can be performed with high success rates and a high degree of safety at high volume centers.
PMCID: PMC3806015  PMID: 24070089
spine, tumor, preoperative embolization, surgery
9.  Pure Spinal Epidural Cavernous Hemangioma with Intralesional Hemorrhage: A Rare Cause of Thoracic Myelopathy 
Korean Journal of Spine  2014;11(2):85-88.
Although cavernous hemangiomas occur frequently in the intracranial structures, they are rare in the spine. Most of spinal hemangiomas are vertebral origin and "pure" epidural hemangiomas not originating from the vertebral bone are very rare. Our spinal hemangioma case is extremely rare because of its "pure" epidural involvement and intralesional hemorrhage. A 64-year-old man presented with progressive paraparesis from two months ago. His motor weakness was rated as grade 4/5 in bilateral lower extremities. He also complained of decreased sensation below the T4 sensory dermatome, which continuously progressed to the higher dermatome level. Magnetic resonance imaging demonstrated thoracic spinal tumor at T3-T4 level. The tumor was located epidural space compressing thoracic spinal cord ventrally. The tumor was not involved with the thoracic vertebral bone. We performed T3-5 laminectomy and removed the tumor completely. The tumor was not infiltrating into intradural space or vertebral bone. The histopathologic study confirmed the epidural tumor as cavernous hemangioma. Postoperatively, his weakness improved gradually. Four months later, his paraparesis recovered completely. Here, we present a case of pure spinal epidural cavernous hemangioma, which has intralesional hemorrhage. We believe cavernous hemangioma should be included in the differential diagnosis of the spinal epidural tumors.
doi:10.14245/kjs.2014.11.2.85
PMCID: PMC4124926  PMID: 25110490
Cavernous; Epidural; Hemangioma; Hemorrhage; Spine
10.  Preoperative Sclerotherapy Using Sodium Tetradecyl Sulphate (Fibro-Vein™) Can Assist in the Management of Vertebral Hemangiomas 
Global Spine Journal  2012;2(3):169-174.
Vertebral hemangiomas are benign lesions accounting for 2 to 3% of all spinal tumors. They are usually asymptomatic and found incidentally on imaging. Uncommonly, vertebral hemangiomas with significant epidural extension can result in radiculopathy or spinal cord compression. Decompressive surgery with or without stabilization is often required when neurological deficits are present. However, surgery can be associated with massive hemorrhage as these tumors are hypervascular. Preoperative embolization and sclerotherapy are well-known management strategies used to minimize intraoperative bleeding and improve symptoms. Recently, the use of sclerosants such as ethanol has decreased, due to reported complications such as Brown–Sequard syndrome. We describe the use of sodium tetradecyl sulfate (Fibro-Vein™, STD Pharmaceutical, Hereford, UK) as an effective alternative to ethanol in the preoperative management of vertebral hemangiomas. To our knowledge, this has not been previously reported. In three patients, we demonstrated minimal intraoperative blood loss using a combination of preoperative embolization of arterial feeders and sclerotherapy with sodium tetradecyl sulfate to control and secure venous drainage. No patients developed complications related to the procedure. In addition to minimal blood loss, a clear dissection plane was also noted intraoperatively.
doi:10.1055/s-0032-1315451
PMCID: PMC3864480  PMID: 24353964
vertebral hemangioma; sclerotherapy; Fibro-Vein™
11.  A 58-Year-Old Woman with Recurrent Hemoptysis after Successful Bronchial Artery Embolization 
Tanaffos  2014;13(1):57-60.
Massive hemoptysis is a life-threatening complication of respiratory disease. It is an emergency requiring immediate medical attention. A 58 year-old woman with bronchiectasis was admitted to the hospital following episodes of massive hemoptysis. Chest CT scan and bronchoscopy did not reveal any endobronchial lesion and bronchial artery angiography and embolization were performed successfully. Despite successful embolization, her hemoptysis recurred and the patient underwent angiography for the 2nd time; which showed normal left bronchial artery and occluded right intercostobronchial artery. Lower thoracic aortogram revealed a systemic non-bronchial artery in the right lower lung field and evidence of pulmonary shunting. Super-selective angiogram of this artery showed vascularity to lower esophagus and considerable supply of the right lower lung field with pulmonary vascular shunting. Embolization of this non-bronchial systemic artery was carried out successfully with complete occlusion. Few days after the embolization, the patient reported pleuritic and epigastric pain and also complained of odynophagia and dysphagia; which were managed conservatively. Four days later, her symptoms improved and she was discharged subsequently. At 40-day follow up, she was still symptom-free with no hemoptysis.
PMCID: PMC4153271  PMID: 25191496
Hemoptysis; Bronchial artery; Embolization
12.  Intramedullary Spinal Cord Metastasis of Choriocarcinoma 
The authors describe a case of choriocarcinoma that metastasized to the cerebral cortex, vertebral body, and intramedullary spinal cord. A 21-year-old woman presented with sudden headache, vomiting and a visual field defect. Brain computed tomography and magnetic resonance examinations revealed an intracranial hemorrhage in the left temporo-parietal lobe and two enhancing nodules in the left temporal and right frontal lobe. After several days, the size of the hemorrhage increased, and a new hemorrhage was identified in the right frontal lobe. The hematoma and enhancing mass in the left temporo-parietal lobe were surgically removed. Choriocarcinoma was diagnosed after histological examination. At 6 days after the operation, her consciousness had worsened and she was in a state of stupor. The size of the hematoma in the right frontal lobe was enlarged. We performed an emergency operation to remove the hematoma and enhancing mass. Her mental status recovered slowly. Two months thereafter, she complained of paraplegia with sensory loss below the nipples. Whole spine magnetic resonance imaging revealed a well-enhancing mass in the thoracic intramedullary spinal cord and L2 vertebral body. Despite chemotherapy and radiotherapy, the patient died 13 months after the diagnosis.
doi:10.3340/jkns.2012.51.3.141
PMCID: PMC3358599  PMID: 22639709
Choriocarcinoma; Intracerebral hematoma; Intramedullary spinal cord metastasis; Spinal metastasis
13.  Unusual presentation of giant cell tumor originating from a facet joint of the thoracic spine in a child: a case report and review of the literature 
Introduction
Giant cell tumor of the synovium is a common benign lesion that frequently occurs at the tendon sheaths in the hand; it is usually found in adults over 30 years old. It is related to pigmented villonodular synovitis. Giant cell tumor of the synovium or pigmented villonodular synovitis has been described rarely in the axial skeleton especially in the thoracic vertebrae of a child.
Case presentation
A previously healthy 7-year-old Thai girl presented with back pain and progressive paraparesis and was unable to walk for 1 month. She had weakness and hyperreflexia of both lower extremities. Magnetic resonance imaging showed a well-defined homogeneously and intensely enhanced extradural mass with cord compression at T4 to T7 levels. The patient underwent laminectomy at T4 through to T7 and total tumor removal. Permanent histopathologic sections and immunostains revealed a giant cell tumor of the synovium. Postoperative neurological status recovered to grade V. Magnetic resonance imaging at the 1-year follow-up showed no recurrence and there was no clinical recurrence at the 2-year follow-up.
Conclusion
We report an extremely rare case of giant cell tumor in the epidural space that extended from a thoracic facet joint. The tumor was removed successfully through laminectomies. Although giant cell tumor of a facet joint of the thoracic spine is very rare, it must be considered in the differential diagnosis for masses occurring in the epidural space in a child. Total tumor removal is the best treatment. Careful monitoring of recurrence can achieve a good clinical outcome.
doi:10.1186/1752-1947-7-178
PMCID: PMC3711725  PMID: 23830026
Giant cell tumor; Spine; Synovium
14.  Endometrial stromal sarcoma metastasis to the lumbar spine and sphenoid bone 
Rare Tumors  2011;3(3):e27.
Endometrial stromal sarcoma (ESS) is typically associated with metastasis to the abdomen, pelvis, and lung. We found three case reports of ESS metastasis to the bone (two to the thoracic spine, and one to the parietal bone). Our objective is to review the literature on ESS spinal and intracranial metastases and, report the first case of ESS metastatic to the lumbar paraspinal region and sphenoid bone. A 53-year-old female with ESS status-post radiation, chemotherapy, and pelvic exenteration surgery presented with right hip weakness, back pain, and radicular leg pain that were explained by chemotherapy-induced neuropathy, radiation-induced lumbosacral plexopathy, and femoral nerve and obturator nerve injury during pelvic exenteration surgery. During routine positron emission tomography, we found metastasis to the L3 lumbar spinal region. L3 laminectomy and subtotal resection of the mass was performed with tumor residual in the neuroforamina and pedicles. One month later, magnetic resonance imaging (MRI) performed for persistent headaches revealed a large lesion in the sphenoid bone that was biopsied transsphenoidally with the same diagnosis, but no further surgery was performed. She is intolerant of chemotherapy and currently undergoing whole brain radiation. Delay in the diagnosis and management of lumbar paraspinal and sphenoid bone metastasis of ESS likely occurred because of the uniqueness of the location and aggressiveness of ESS metastasis. Health care providers should be aware of potentially aggressive metastasis of ESS to bone, in particular the unusual locations of the lumbar paraspinal region and sphenoid bone.
doi:10.4081/rt.2011.e27
PMCID: PMC3208414  PMID: 22066034
endometrial stromal sarcoma; uterine sarcoma; lumbar spine; sphenoid bone; bone metastasis.
15.  Non-Hodgkin's lymphoma “masquerading” as Pott's disease in a 13-year old boy 
Lymphomas are malignant neoplasms of the lymphoid lineage. They are broadly classified as either Hodgkin disease or as non-Hodgkin lymphoma (NHL). Burkitt's lymphoma, a variety of NHL, is significantly most common in sub-Saharan Africa, where it accounts for approximately one half of childhood cancers. Lymphoblastic lymphoma is less common. A case of paravertebral high grade non-Hodgkin's lymphoma (lymphoblastic lymphoma) “masquerading” as Pott's disease in a 13-year-old child is reported. The present report was informed by the unusual presentation of this case and the intent of increasing the index of diagnostic suspicion. A brief appraisal is provided of the clinical parameters, management strategies and challenges. AT was a 13-year boy that presented on account of a slowly evolving and progressively increasing hunch on the back and inability to walk over 4 and 8 months duration, respectively. There was subsequent inability to control defecation and urination. There was no history of cough. He and his twin brother lived with their paternal grandfather who had chronic cough with associated weight loss. The grandfather died shortly before the child's admission. The child had no BCG immunization. The essential findings on examination were in keeping with lower motor neurons (LMN) paralysis of the lower limbs. The upper limbs appeared normal. There was loss of cutaneous sensation from the umbilicus (T10) downward. There was a firm, (rather tense), non-tender non-pulsatile, smooth swelling over the mid-third of the back (T6-L1) the mass had no differential warmth. It measures about 20×12 cm. Chest radiograph showed no active focal lung lesion, but the thoraco-lumbar spine showed a vertebral planner at L1 and a wedged collapse of T11-T12 vertebrae. There was sclerosis of the end plates of all the vertebral bodies with associated reduction in the bone density. He had an excision biopsy on the 90th day on admission, following which his clinical state rapidly deteriorated. He died within 48 h post surgery. This report aims at raising the local index of clinical suspicion by highlighting the reality of rarities, even in the presence of inadequate diagnostic facilities!
doi:10.4103/0971-5851.89793
PMCID: PMC3237173  PMID: 22174499
Non-Hodgkin's lymphoma; lyphoblastic lymphoma; Pott's disease
16.  Lumbar Spinal Extradural Angiolipoma: Case Report and Review of the Literature 
Angiolipomas in the lumbar spinal region are extremely rare. The present report describes the identification of such a tumor and its removal, and discusses the tumor characteristics and prognosis. A 74-year-old woman was presented with a 5-month history of lower back pain. Severe radiculopathy was experienced in the left leg for 5 days prior to the presentation, and there were no neurological deficits. Magnetic resonance (MR) images showed an approximately 3.5 cm heterogeneously enhanced and elongated mass at the left L5-S1 level. A portion of the mass appeared with high signal intensity on T2-weighted MR images, with low signal intensity on T1-weighted images, and with high signal intensity on T1 fat suppression enhancement images. Resection of the tumor was approached via an L5 and S1 laminectomy. A fibrous sticky yellowish hypervascular tumor was identified. Histological study revealed the tumor as an angiolipoma. Symptoms were relieved after tumor excision, and there were no neurological sequelae. Although extremely rare, lumbar epidural angiolipoma should be considered in the differential diagnosis of lumbar spinal epidural lesions. The prognosis after surgical management of this lesion is favorable.
doi:10.3340/jkns.2008.44.4.265
PMCID: PMC2588319  PMID: 19096690
Angiolipoma; Spinal neoplasm; Lumbar epidural tumor
17.  Cardiovascular changes after PMMA vertebroplasty in sheep: the effect of bone marrow removal using pulsed jet-lavage 
European Spine Journal  2010;19(11):1913-1920.
Clinically, the displacement of intravertebral fat into the circulation during vertebroplasty is reported to lead to problems in elderly patients and can represent a serious complication, especially when multiple levels have to be treated. An in vitro study has shown the feasibility of removing intravertebral fat by pulsed jet-lavage prior to vertebroplasty, potentially reducing the embolization of bone marrow fat from the vertebral bodies and alleviating the cardiovascular changes elicited by pulmonary fat embolism. In this in vivo study, percutaneous vertebroplasty using polymethylmethacrylate (PMMA) was performed in three lumbar vertebrae of 11 sheep. In six sheep (lavage group), pulsed jet-lavage was performed prior to injection of PMMA compared to the control group of five sheep receiving only PMMA vertebroplasty. Invasive recording of blood pressures was performed continuously until 60 min after the last injection. Cardiac output and arterial blood gas parameters were measured at selected time points. Post mortem, the injected cement volume was measured using CT and lung biopsies were processed for assessment of intravascular fat. Pulsed jet-lavage was feasible in the in vivo setting. In the control group, the injection of PMMA resulted in pulmonary fat embolism and a sudden and significant increase in mean pulmonary arterial pressure. Pulsed jet-lavage prevented any cardiovascular changes and significantly reduced the severity of bone marrow fat embolization. Even though significantly more cement had been injected into the lavaged vertebral bodies, significantly fewer intravascular fat emboli were identified in the lung tissue. Pulsed jet-lavage prevented the cardiovascular complications after PMMA vertebroplasty in sheep and alleviated the severity of pulmonary fat embolism.
doi:10.1007/s00586-010-1555-y
PMCID: PMC2989274  PMID: 20725752
Vertebroplasty; Pulmonary fat embolism; Cardiovascular changes; Pulsed jet-lavage; In vivo study
18.  Management of Refractory Metastatic Anal Squamous Cell Carcinoma Following Disease Progression on Traditional Chemoradiation Therapy 
Case Study
Ms. S.G., a 56-year-old woman with a poorly differentiated squamous cell carcinoma of the anal canal, American Joint Committee on Cancer stage III (T2, N1, M0), was initially diagnosed in December, 2007 at an outside institution after she had noted blood in her stool for approximately 6 months. Her medical history was unremarkable. She had no known history of HIV or other sexually transmitted diseases. At the time of presentation, Ms. S.G. had an Eastern Cooperative Oncology Group performance status of 1 related to cancer-related pain. Her appetite and weight were both stable.
A complete colonoscopy demonstrated a large, immobile, ulcerated, firm, 4-cm lesion in the distal rectum, arising from the anal canal. Initial staging positron emission tomography/computed tomography (PET/CT) scan revealed a hypermetabolic inferior anorectal mass with left perirectal and presacral nodal metastases. There was no definite evidence of distant metastatic disease.
Ms. S.G. received chemoradiation treatment following her diagnostic studies, with a total dose of 45 Gy over 26 fractions to the pelvis with concurrent infusional fluorouracil (5-FU; 2, 450 mg over 7 days) and mitomycin C (12 mg/m2 on day 1) at an outside institution. However, during her chemoradiation therapy, Ms. S.G. experienced a 3-week treatment break due to severe radiation dermatitis, as recommended by her outside treating oncologist.
Upon treatment completion, Ms. S.G. underwent a biopsy of the anal canal, which revealed no evidence of residual malignancy. As recommended by her treating oncologist, she received four additional cycles of adjuvant infusional 5-FU in combination with leucovorin. Shortly thereafter, Ms. S.G. developed progressive pelvic pain. She underwent a second PET/CT scan, revealing mixed findings: interval resolution of abnormal standardized uptake value (SUV) activity at the primary tumor in the anal canal, but an increase in the size and SUV of nodal disease within the left perirectal and presacral regions. A CT-guided biopsy noted a perirectal abscess requiring drainage but was inconclusive for disease recurrence; Ms. S.G. was treated with IV antibiotics.
Six weeks later, repeat radiographic imaging noted additional changes suspicious for regional recurrence, which was biopsy-confirmed. Ms. S.G. was subsequently referred to MD Anderson Cancer Center for consideration of salvage pelvic exenteration.
On physical exam a mass was palpated in the left lower quadrant, but there was no evidence of inguinal adenopathy. On digital rectal exam there was notable external erythema with a fixed mass and moderate sphincter tone. A chest CT scan showed no definite evidence of metastatic disease, but an MRI of the abdomen/pelvis indicated the presence of a complex partially necrotic mass (7.6 × 4.9 × 7.3 cm3) extending to the rectosigmoid junction, inseparable from the left lateral bowel wall, with partial encasement of the bowel. In addition, there was infiltration of the left piriformis muscle and cervix consistent with local recurrence. She was referred to medical oncology and radiation oncology for consideration of reirradiation with concurrent neoadjuvant chemotherapy for palliation and possible surgical resection.
In early December 2008, Ms. S.G. received intensity-modulated radiation therapy (IMRT), with a total dose of 27 Gy over 18 fractions. She received concurrent infusional 5-FU at 300 mg/m2/day, from Monday to Friday, on the days of radiation. She also received a weekly bolus dose of cisplatin at 20 mg/m2. The intent was to treat to 30 Gy, but the patient deferred further treatment early due to anorectal irritation. She then underwent restaging with a PET/CT scan and a pelvic MRI in February 2009, revealing radiographic partial response of the known pelvic recurrence and reduced pelvic pain (Figures 1A and 1B). Figure 1 Figure 1. Contrast-enhanced axial MRI image of the lower pelvis. (A) Pretreatment, complex mass at the rectosigmoid junction measuring approximately 7.6 × 4.9 × 7.3 cm3. (B) Posttreatment, large necrotic mass measuring 3–4 cm in greatest dimension.
Unfortunately, in the interim, she developed multiple bilateral liver lesions and punctate pulmonary nodules consistent with distant disease (Figures 2A, 2B, and 3A). Figure 2 Figure 2. Contrast-enhanced axial CT images of the lung. (A) Subcentimeter nodular opacity in the left upper lobe. (B) Subcentimeter opacity in the right upper lung lobe. Figure 3 Figure 3. Contrast-enhanced axial CT image of the liver. (A) Pretreatment, multiple bilateral liver lesions. (B) Posttreatment, near-complete resolution of liver lesions.
Ms. S.G. proceeded to undergo systemic chemotherapy with carboplatin at an area under the concentration-time curve of 5 and paclitaxel at 175 mg/m2 day 1, every 21 days. She tolerated the treatment well. After three cycles of chemotherapy, radiographic imaging indicated a mixed response to treatment: interval resolution of the pulmonary nodules, stability of disease in the pelvic mass, but progression of the hepatic metastases.
Given Ms. S.G.'s continuing excellent performance status, further treatment was recommended. Based on recent published literature, a regimen of cisplatin at 80 mg/m2 day 1, vinorelbine at 25 mg/m2 day 1 (repeated every 28 days), and weekly cetuximab (VCC) at 250 mg/m2 was initiated. Remarkably, following three cycles of treatment, despite receiving multiple prior lines of chemotherapy, her restaging CT scan demonstrated complete radiographic response of the intrathoracic disease, stable response of the anorectal mass, and near-complete resolution of the hepatic lesions (Figures 3A and 3B).
Overall, Ms. S.G. had tolerated her treatment very well. Given her response and tolerability, she was evaluated again for curative surgical resection. However, she opted to receive the VCC regimen closer to home and was lost to follow-up. Unfortunately, we were unable to obtain medical records confirming if she indeed received additional treatment as recommended. Ms. S.G. was noted to have passed away due to progression of her disease approximately 6 months later.
PMCID: PMC4093318  PMID: 25031942
19.  The Fracture Unit Model. A Model for Implementation in Italy: “Multidisciplinary Approach for the Prevention and Treatment of Osteoporotic Vertebral Compression Fractures: VCF Unit” 
Reduced BMD is a risk factor for vertebral fractures (VFs). Every one SD increase in BMD is associated with a 2- to 2.5-fold increase in the risk of VFs. The presence of a previous fracture, vertebral or of other districts, is another important predictor of an increased risk of future fractures, independently of the association between BMD and fracture risk. Thus, the presence of both a low BMD and a previous fracture dramatically increases fracture risk. The definition of osteopo-rotic VFs has undergone considerable variations over the years, going from initial clinical sign of OP, through the now superseded definition of VFs as a disease, to a complication of OP resulting from bone fragility. The prevalence of VFs increases with age in both sexes, and it is calculated that at the age of 80 years, 37% of Caucasian women will have at least one radiographically evident VF. It has been estimated, again in Caucasian women, that the percentage incidence of fractures is 0.5% in those aged 50-55 years, 1.4% in those aged 65-69 years, and over 2% in women older than 75. However, two factors prevent an accurate assessment of the epidemiology of VFs. First, most VFs escape clinical diagnosis. Second, the absence of a “gold standard” radiographic definition of VFs has given rise to different ways of defining these lesions. VFs are rarely a cause of mortality, but they are associated with increased impairment of general conditions. Recurrent VFs have irreversible clinical consequences, such as reduction of height and chronic vertebral pain, which provoke an intensification of the pain and a greater degree of disability due to accentuation of kyphosis. The presence of VFs and kyphosis leads to a reduced thoracic volume and, consequently, to a loss of lung volume, in some cases severe enough to result in respiratory insufficiency. The consequences of the intense pain are: reduced range of motion, loss of balance, slowed gait and greater difficulty carrying out normal daily activities. In rare cases, lower limb pain and weakness may appear, caused by compression of the spinal medulla by the deformed vertebral body. The main aim of treatment is to restore the patient to his/her pre-trauma levels of functioning. This can be achieved through recourse to mini-invasive percutaneous techniques, vertebroplasty and kyphoplasty, with the aim of reducing the pain caused by osteo-porotic vertebral compression fractures, of preventing progression of the vertebral collapse and of rapidly re-establishing functional activity. Most fracture patients are discharged without undergoing a thorough bone metabolism assessment that could identify the causal factor of the fracture. In a high percentage (up to 95%) of patients with recent fractures, BMD is not measured and, therefore, a diagnosis of OP is not made. Consequently, these patients are not prescribed drugs capable of effectively reducing the risk of further fractures. Specialist orthopaedic centres need to introduce protocols designed to ensure application of the current procedures for diagnosing and treating OP.
On the basis of these considerations, we undertook to develop, in collaboration with the Department of Specialist Surgical Sciences of the University of Florence, the Orthopaedics and Traumatology Units 1, 2 and 3, the Recovery and Functional Re-education Unit, the Neurosurgery Unit, and the third Radiodiagnostics Unit of the Careggi Hospital in Floren-ce, a protocol that involves a range of specialists in assessing the introduction of variable, outcome-targeted medical therapies for osteoporotic patients submitted to kyphoplasty following fragility fractures of the vertebra. To choose the appropriate medical therapy, and to monitor its effects, the patients will be submitted to a series of clinical investigations. The “appropriate” therapy could include calcium and vitamin D supplementation, biphosphonates, SERMs, bone anabolic agents and combinations of drugs. The safety of the medical therapy and any adverse effects will be monitored at each follow-up visit through an appropriate questionnaire. This study aims to compare the outcomes of the group following a traditional pathway with those following a modified pathway (prescription of a targeted medical therapy), by means of metabolic, instrumental and functional tests performed at 2 months, 6 months, 1 year and 2 years. The general aim of the study will be to evaluate the efficacy and safety of a modified versus a traditional pathway in the care of osteoporotic patients undergoing kyphoplasty for VFs. The primary endpoint of the study will be the percentage of successes in the modified compared with the traditional pathway group. Secondary endpoints will be: change in femoral and lumbar BMD, changes in biochemical markers of bone remodelling and quality of life, assessment of safety parameters: overall and symptomatic cement leakage, pulmonary embolism, spinal medulla compression, radicular pain, radiculopathies and assessment of total procedure-related, cement-related and access-related adverse events. The ultimate aim of the study will be to prepare guidelines for the management, in terms of metabolic diagnosis and relative medical therapy, of patients with OP complicated by VFs.
PMCID: PMC3213825
20.  Mediastinal Choriocarcinoma Masquerading as Relapsed Hodgkin Lymphoma 
Case Reports in Oncology  2011;4(3):512-516.
Primary mediastinal choriocarcinoma is a rare extragonadal germ cell malignancy. We describe the first case of a patient who developed mediastinal choriocarcinoma after treatment for Hodgkin lymphoma (HL). A 25-year-old man with classic HL, nodular sclerosis subtype, underwent treatment with splenectomy followed by radiation therapy. Unfortunately, his disease relapsed with a paraspinal mass, and he was subsequently treated with MOPP (mechlorethamine, Oncovin, procarbazine, and prednisone) alternating with ABVD (Adriamycin, bleomycin, vinblastine, and dacarbazine). He achieved a complete remission after 6 cycles. Ten years after treatment, the patient presented with a persistent cough, haemoptysis, right supraclavicular lymphadenopathy, and weight loss. His chest X-ray showed opacification of the lower right hemithorax with a widened mediastinum. Given unresponsiveness to several antibiotics and lack of evidence for lung volume loss, there were concerns over lung infiltration with relapsed lymphoma. Transbronchial fine needle aspiration biopsy suggested recurrence of his HL. MOPP alternating with ABVD was again given. Due to disease progression, brachytherapy as well as a cocktail of dexamethasone, cytarabine, and cisplatin were also tried. However, on a subsequent excisional lymph node biopsy, it turned out that the tumour was in fact choriocarcinoma and not relapsed HL. Unfortunately, despite aggressive therapy, the patient's disease rapidly progressed, and he died within 2 weeks.
doi:10.1159/000334080
PMCID: PMC3220912  PMID: 22114578
Germ cell tumour; Choriocarcinoma; Hodgkin lymphoma; Mediastinal tumour
21.  Tailored Surgical Approaches for Benign Craniovertebral Junction Tumors 
Objective
We report our surgical experience in the treatment of 16 consecutive patients with benign craniovertebral junction (CVJ) tumor, observed from 2003 to 2008 at our department.
Methods
We had treated 6 foramen magnum meningiomas, 6 cervicomedullary hemangioblastomas, 1 accessory nerve schwannoma, 1 hypoglossal nerve schwannoma, 1 C2 root schwannoma, and 1 cavernous hemangioma. Clinical results were evaluated by Karnofsky Performance Scale (KPS) and all patients underwent preoperative neuroradiological evaluation with computed tomography (CT) and magnetic resonance image (MRI). Angiography was performed in 15 patients and preoperative embolization was done in 2 patients.
Results
Five far-lateral, 1 supracondylar and 10 midline suboccipital approaches were performed. Gross total removal was achieved in 15 cases (94%) and subtotal removal in 1 patient (6%). None of the patients required occipitocervical fusion. Radiological follow-up showed no recurrence in cases totally removed. Postoperative decrease of KPS scores was recorded in only 1 patient. The treatment of cervicomedullary solid hemangioblastoma presented particular issues : by preoperative embolization, we removed tumor totally without an excessive bleeding or brainstem injury. In one of foramen magnum meningioma, we carried out subtotal removal due to hard tumor consistency and encasement of neurovascular structures.
Conclusion
: The choice of surgical approaches and the extent of bone resection should be defined according to the location and size of individual tumors. Moreover, we emphasize that preoperative neuroradiological evaluations on presumptive tumor type could be helpful to the surgeon in tailoring the technique and providing the required exposure for different lesions, without unnecessary surgical steps.
doi:10.3340/jkns.2010.48.2.139
PMCID: PMC2941857  PMID: 20856663
Benign tumor; Complication; Craniovertebral junction; Neuroradiology; Surgical resection
22.  Postoperative spinal epidural hematoma resulting in cauda equina syndrome: a case report and review of the literature 
Cases Journal  2009;2:8584.
Spinal epidural hematoma is a well known complication of spinal surgery. Clinically insignificant small epidural hematomas develop in most spinal surgeries following laminectomy. However, the incidence of clinically significant postoperative spinal epidural hematomas that result in neurological deficits is extremely rare. In this report, we present a 33-year-old female patient whose spinal surgery resulted in postoperative spinal epidural hematoma. She was diagnosed with lumbar disc disease and underwent hemipartial lumbar laminectomy and discectomy. After twelve hours postoperation, her neurologic status deteriorated and cauda equina syndrome with acute spinal epidural hematoma was identified. She was immediately treated with surgical decompression and evacuation of the hematoma. The incidence of epidural hematoma after spinal surgery is rare, but very serious complication. Spinal epidural hematomas can cause significant spinal cord and cauda equina compression, requiring surgical intervention. Once diagnosed, the patient should immediately undergo emergency surgical exploration and evacuation of the hematoma.
doi:10.4076/1757-1626-2-8584
PMCID: PMC2740261  PMID: 19830087
23.  Caesarean scar choriocarcinoma: a case report and review of the literature 
Objective
To report the clinical characteristics, pathologic findings and treatments of a patient with a Caesarean scar choriocarcinoma.
Patient history
A 22-year-old woman had a diagnosis of primary gestational choriocarcinoma in a uterine Caesarean scar misdiagnosed as a normal Caesarean scar pregnancy. The patient underwent selective uterine artery embolization coupled with methotrexate arterial injection, along with dilatation and curettage of the uterine Caesarean scar. Finally, she received eight courses of multiagent chemotherapy. The reproductive function of the patient was preserved.
Conclusions
Primary gestational choriocarcinoma out of the uterine corpus is a rare disease. A Caesarean scar choriocarcinoma is an extremely unusual example of this entity because of its unique position. To the best of our knowledge, this is the first report of this phenomenon. Our experience and a literature review suggest that a clinical diagnosis of a primary gestational choriocarcinoma of the uterine Caesarean scar is difficult to make, and uterine artery embolization is beneficial to prevent massive bleeding before curettage.
doi:10.1186/2047-783X-19-25
PMCID: PMC4026121  PMID: 24887563
Caesarean scar; Choriocarcinoma; Treatment
24.  Intracranial hemorrhage following lumbar spine surgery 
European Spine Journal  2012;21(10):2091-2096.
Background
Intracranial hemorrhage is a serious but rare complication of spinal surgery, which can occur in the intracerebral, cerebellar, epidural, or subdural compartment.
Purpose
To describe patients with intracranial hemorrhage after lumbar spinal surgery and present clinical and diagnostic imaging findings.
Methods
In this retrospective study, medical records of 1,077 patients who underwent lumbar spinal surgery in our tertiary referral neurosurgery center between January 2003 and September 2010 were studied. The original presentations of the patients before the surgical intervention were herniated lumbar disc, spinal canal stenosis, spondylolisthesis, lumbar spinal trauma, and lumbar spine and epidural tumor. The operations performed consisted of discectomy, multiple level laminectomy, stabilization and fusion, lumbar instrumentation, and lumbar spinal and epidural tumor resection.
Results
Four cases developed intracranial hemorrhage including acute subdural hematoma (one case), epidural hematoma (one case), and remote cerebellar hemorrhage (two cases). The clinical and diagnostic imaging characteristics along with treatments performed and outcomes of these four patients are described and the pertinent literature regarding post-lumbar spinal surgery intracranial hemorrhages is reviewed.
Conclusion
Though rare, intracranial hemorrhage can occur following lumbar spinal surgery. This complication may be asymptomatic or manifest with intense headache at early stages any time during the first week after surgery. Dural tear, bloody CSF leakage, focal neurologic symptoms, and headache are indicators of potential intracranial hemorrhage, which should be considered during or following surgery and necessitate diagnostic imaging.
doi:10.1007/s00586-012-2187-1
PMCID: PMC3463703  PMID: 22349967
Intracranial hemorrhage; Lumbar spine surgery; Remote cerebellar hemorrhage; Subdural hematoma; Epidural hematoma
25.  Cutaneous Manifestation of Metastatic Infantile Choriocarcinoma 
Case Reports in Pediatrics  2014;2014:104652.
Infantile choriocarcinoma is a highly malignant rare germ cell tumor that arises from the placenta. Simultaneous intraplacental choriocarcinoma involving both mother and infant is extremely rare. Cutaneous metastasis in infantile choriocarcinoma is even rarer with only a few case reports available. Here we describe a case of a female neonate who presented to the ED with a rapidly growing and bleeding vascular lesion to her right cheek. She was eventually diagnosed by biopsy with metastatic choriocarcinoma. In addition to the cutaneous tumor, she also had metastatic disease in her lungs. Her mother was subsequently found to have choriocarcinoma with metastatic disease to the lungs as well.
doi:10.1155/2014/104652
PMCID: PMC4145388  PMID: 25197602

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