Intradural hydatid cysts of the spinal cord

A patient with primary intradural spinal hydatid disease presenting with complete paraplegia is described. The diagnosis was made at operation. The patient did not have any clinical evidence of visceral or extradural hydatid lesions. This, we believe, is the first case of primary spinal intradural hydatid disease reported from India.     

脊柱包虫病的诊断与治疗

目的:探讨脊柱包虫病的诊断及治疗方法。方法:1990年10月～2010年10月手术治疗脊柱包虫病患者9例,病程为3个月～12年。临床表现为午后低热、腰背疼痛和腰背部包块,术前脊髓神经功能Frankel分级A级1例,B级2例,C级2例,D级4例。包虫病8项免疫试验均为阳性。术前均行影像学检查,病变单纯累及胸椎2例(T8、T9 1例,T11、T12 1例),腰椎2例(L3 1例,L1、L2、L3 1例),胸腰段4例(T12、L1 2例,T12、L1、L2 2例),骶椎1例。X线片、CT检查误诊为椎体结核5例、转移瘤2例、脊索瘤1例、腰大肌脓肿1例。MRI检查7例诊断为脊柱包虫病;2例病变单纯累及腰椎者缺乏囊中囊典型信号改变,误诊为脊柱转移瘤。均行椎管减压病灶清除植骨内固定术,其中病变单纯累及胸椎的2例与骶骨1例行后路全椎板切除减压,2例腰椎病变者行椎板间开窗椎管减压,4例胸腰段病变者行脊柱侧前方入路经横突椎管减压。7例术前诊断脊柱包虫病者术前口服抗包虫药阿苯达唑2～3周,所有患者术后继续服用阿苯达唑3个月。结果:手术时间210～330min,平均260min;术中失血量170～470ml,平均300ml。7例术前MRI检查诊断为脊柱包虫病者术后病理诊断为脊柱细粒棘球蚴病,2例术前MRI误诊为脊柱转移瘤者术后病理诊断为腰椎泡状棘球蚴病。随访1～10年,平均4.6年。1例腰椎泡状棘球蚴病患者术后8个月植骨未愈合,经理疗、促骨生长药物治疗,1年后复查X线片显示骨性愈合;其余8例患者植骨区骨性融合,愈合时间为5～8.5个月,平均7个月。末次随访患者脊髓神经功能Frankel分级改善6例,无变化3例。随访期间无复发。结论:MRI检查对脊柱细粒棘球蚴病有诊断价值,对腰椎泡状棘球蚴病易误诊,后者的确诊有赖于病理检查;脊柱包虫病在药物治疗的基础上采用手术治疗可取得较好疗效。     

Primary spinal intradural extramedullary hydatid cyst in a child

BACKGROUND/OBJECTIVE: Spinal hydatid cyst is a serious form of hydatid disease affecting less than 1% of the total cases of hydatid disease. We present a case of pathologically confirmed primary intradural spinal cyst hydatid in an otherwise healthy patient who showed no other evidence of systemic hydatid cyst disease. CASE REPORT: An 8-year-old boy presented with back pain, left leg pain, and difficulty in walking. The patient had no other signs of systemic hydatid cyst disease. An intradural extramedullary cystic lesion was identified with magnetic resonance imaging and was shown to be a hydatid cyst by histopathologic examination after the surgical removal. CONCLUSION: Although extremely rare, primary intradural extramedullary hydatid cyst pathology might be the cause of leg pain and gait disturbance in children living in endemic areas.     

Diagnostic pitfalls of spinal echinococcosis

OBJECTIVE: To critically examine and elucidate the diagnostic pitfalls of spinal echinococcosis. SUMMARY OF PATIENTS: From October 1957 to June 2006, 25 consecutive cases drawn from 5721 cases of echinococcosis were collected in the First Affiliated Hospital of Xinjiang Medical University. The selected cases comprised 11 males and 14 females; all were treated with debridement operations. The average age was 28.3 years (15 to 56 y). The average duration of infestation with spinal hydatid disease was 3.2 years (0.5 to 12 y). Nineteen of the 25 cases underwent magnetic resonance imaging (MRI) scanning, which identified 17 out of 19 cases as having hydatid disease. The lesion was located in the cervical vertebrae in 3, the thoracic vertebrae in 11, the lumbar vertebrae in 5, and the sacrum in 6 cases. RESULTS: Eighteen cases were available for follow up; the period ranging from 0.5 to 15 years with an average of 3.6 years. The Casoni test was performed in 15 cases and was positive in 12 patients (80%). In addition, 4 cases were positive in all of the so-called 8 tests of immunodiagnostic methods. MRI examination was performed in 19 of the 25 cases and 17 of these were diagnosed as having spinal hydatid disease (89.47%). The typical MRI appearance is that of a multilocular cyst and the signal of the parent cyst is similar to that of muscle and higher than that of secondary cyst in the T1Weighted image (WI). The signal of the secondary cyst is similar to water, either located in or overflowing or adjacent to the parent cyst. Both the parent and the secondary cysts showed high signals in the TW1 with either rose or wheel shapes. In the 18 cases, which were reviewed, 11 cases had relapsed (61.11%). CONCLUSIONS: Although x-ray or computed tomography images of spinal echinococcosis are similar to tuberculosis, metastases, giant cell tumors, or cysts of the bone, MRI shows distinctive diagnostic features of spinal hydatid disease. Serologic examinations are important to confirm the correct diagnosis.     

Intradural spinal hydatid cysts

Spinal hydatid cysts are very rare and comprise only 1% of all bony involvement. Intradural hydatid cysts are extremely rare compared to other types of spinal hydatid cysts. We report the case of a 19-year-old man with lumbar intradural hydatid cysts. He complained of paraparesis and urinary hesitancy. Myelography revealed a block of the contrast medium at the L4 level and multiple round radiolucent lesions rostrally. At surgery, multiple hydatid intradural cysts were extirpated. The patient’s neurologic deficits improved postoperatively, but recurred 6 weeks later. Reoperation led to partial improvement of deficits. Primary intradural hydatid cysts are extremely rare. It is difficult to explain an isolated intradural location of multiple cysts. In our patient, the cysts were multiple and the patient’s cerebral CT scan was normal; he had undergone no previous lumbar puncture. So we can hypothesize that intradural spinal cysts may be primarily multiple. Received: 7 May 1996 Revised: 9 August 1996 Accepted: 17 August 1996     

Primary spinal extradural hydatid cyst in a child: case report and review of the literature

Spinal hydatid cyst is a rare but serious condition. An 8-year-old boy presented with back pain, progressive weakness and numbness in both legs. Magnetic resonance imaging (MRI) of the lumbar region showed a cystic lesion with regular contour located in extradural space. There was cerebrospinal fluid- (CSF-) like signal intensity on T1- and T2-weighted images. The lesion had excessively compressed the dural sac and caudal roots, and expanded to the L3 and L4 neural foramina. The case was explored with L2, L3, L4 laminectomy and the hydatid cyst was removed totally. The clinical presentation, diagnosis and surgical treatment of this rare case of spinal hydatid disease is discussed, and all available cases of primary extradural hydatid cyst reported in the literature are presented.     

Hydatid cysts in uncommon locations in the same patient: simultaneous cardiac and spinal involvement

INTRODUCTION: The spine and cardiac cavities are uncommon locations for hydatid disease. Spinal and cardiac hydatid cases separately constitute only 0.5-1% of all hydatid cases. We reported a case with spinal and cardiac involvement simultaneously. Clinical and radiological findings of this unique case are discussed. CASE REPORT: A thirty-four-year-old male patient was admitted to hospital with pain at the low back and left thigh. He had undergone surgery for lumbosacral hydatidosis twice before. Cysts had been removed totally in the last operation. The disease relapsed with widespread involvement of the sacropelvic region. During the investigation, we diagnosed a cyst in the cardiac cavity, incidentally. The cyst in the cardiac cavity was removed totally. DISCUSSION: Hydatid disease is still an endemic disease in South America and some Mediterranean countries including Turkey. Cysts rarely involve the spine and cardiac cavities. The simultaneous involvement of the sacropelvic region and the cardiac cavity is an extremely rare condition. Cardiac cysts have a poor prognosis. Prompt surgical extraction of the cyst is a critical mainstay of the management.     

Multiple primary chest wall hydatid cysts associated with spinal canal involvement

Primary multiple chest wall hydatid cysts associated with spinal canal involvement through an intervertebral foramen is an uncommon clinical entity. We present a 54-year-old man who underwent cystotomy and total resection of ribs five through seven via a left posterolateral thoracotomy followed by Th5-Th6 anterolateral partial pediculotomies for removal of cysts in the spinal canal. Although spinal reconstruction was not required, the chest wall defect was repaired with mersilene mesh-methyl methacrylate sandwich graft. Hydatid disease should be considered in the differential diagnosis of mass lesions located in the chest wall. In cases of spinal canal involvement, detailed visualization of spinal canal utilizing MRI and/or CT is essential for planning surgical approach.     

Spinal hydatid disease

STUDY DESIGN: Review article on spinal involvement of hydatid disease. OBJECTIVES: A better understanding of this rare but clinically challenging disease is intended. An overview of the epidemiology, pathogenesis, presentation and diagnosis of spinal hydatid disease is provided. Management problems and frequent pitfalls are discussed as well as current therapeutic options, results and outcome. METHODS: Thirty-seven reports of spinal hydatid disease published between 1964 and 2000 were reviewed. RESULTS: Most of the reported cases of spinal hydatid disease presented with spinal cord compression syndrome. Due to the relative rarity of the problem the diagnosis was frequently made during surgery. Surgical intervention by decompressive laminectomy was the most frequent first management. Reports of anterior procedures as well as spinal stabilization exist. Intraoperative prophylaxis to reduce spillage as well as pharmacotherapy were usually instituted. Results of surgery were generally reported to be poor. Progressive neurological and mechanical deterioration over the years was the most frequently reported disease course. Anecdotal reports of alternative management strategies exist. CONCLUSION: Spinal hydatid disease should be considered in the differential diagnosis of spinal cord compression syndrome in endemic countries and sought after with imaging and serology. Treatment is based on surgical decompression. Despite therapy the disease frequently relapses with progressive destruction of the vertebral column and neurological deterioration. Retention of spinal stability is the major long term concern. Overall outcome is poor with few reports of disease-free long term survival.     

脊柱包虫病的临床特点及诊断方法

目的探讨脊柱包虫病的临床特点,为诊断和治疗提供依据.方法回顾性分析1994年7月至2003年10月诊治的14例脊柱包虫病患者,男5例,女9例;年龄5～23岁,平均25.8岁.患者均有牧区生活史,病程为4～120个月,平均26.7个月.除行常规检查外所有患者均拍摄X线片,8例行CT扫描,13例行MR检查,8例行免疫血清学检查.病变多累及胸椎,占35.8%,其次为腰椎和腰骶椎,分别为21.4%和28.6%,颈椎较少见.根据Dew分类,包虫累及髓外硬膜内1例,椎管内硬膜外2例,脊椎7例,椎旁1例,其余3例累及硬膜外周围组织.所有患者均行手术治疗,并行组织病理学检查.结果脊柱包虫病的临床症状主要表现为病变部位的肿胀、疼痛或放射痛,后逐渐发展为脊髓神经损害.X线检查缺乏特征性,仅表现为局部不规则骨质破坏,可伴边缘硬化或钙化.CT主要表现为多个大小不等的囊状低密度缺损.MRI示病变呈多囊性生长,T1加权像上囊壁与囊内容物均呈低信号,T2加权像上囊壁呈低信号,囊内容物呈高信号,簇集呈“葡萄串样”;合并感染时,T1加权像和T2加权像信号均增强.8例行包虫三项检查的患者中7例阳性,其中3例接受包虫八项检查,均为阳性.组织病理学检查均为细粒棘球绦虫.结论MRI是诊断脊柱包虫病最敏感的方法,在确定病变部位和范围上具重要作用;CT和免疫血清学检查有助于包虫病的诊断和鉴别诊断;提高对该病的认识是防止误诊的关键.     

Primary spinal extradural hydatid cyst

We report a rare case of spinal cord compression caused by a primary extradural hydatid cyst.     

Intradural extramedullary primary hydatid cyst of the spine in a child: a very rare presentation

Spinal hydatid cyst is a serious but fortunately uncommon manifestation of the parasite Echinococcus, involving less than 1% patients with hydatid disease. Intradural hydatid cysts are extremely rare compared to other types of spinal hydatid cysts. We report a rare case of intradural, extramedullary spinal hydatid cyst in a 9-year-old male boy, who presented with weakness of both lower limbs for the last 4 months that was confirmed histopathologically; a better understanding of this rare but clinically challenging disease is intended by reporting this case.     

Effect of albendazole on human hydatid cysts: an ultrastructural study

Five patients with hepatic (3), pelvic (1) or spinal (1) hydatid cysts received 10 mg/kg/d albendazole for 1-3 months prior to surgery. Daughter cysts were present in the spinal hydatid and in one patient with hepatic disease. Electron microscope examination of the cyst tissue of the pelvic and the 2 hepatic cysts lacking daughter cysts showed no evidence of germinal layer, and the protoscoleces were dead. The primary cyst of the hepatic hydatid with daughter cysts (1 month therapy) was also judged dead but some pieces of the daughter cyst germinal layer appeared normal and had unaffected protoscoleces. The daughter cyst tissue of the spinal hydatid (3 month therapy) appeared normal and the protoscoleces viable. In view of the undetermined viability of human hydatids before chemotherapy, treatment of longer than 1 month is advocated for hepatic cysts, particularly if daughter cysts are present, and longer therapy is indicated for spinal disease.     

Diagnosis and treatment of multiple hydatid cysts at the craniovertebral junction. Case report

Echinococcosis, or hydatid disease, of the craniovertebral junction and skull base is rare. The authors report the occurrence of multiple hydatid cysts at this anatomical location in a young woman who was previously misdiagnosed with tuberculosis. The patient underwent transoral excision of the hydatid cysts followed by posterior decompression and occipitocervical fusion. She was treated postoperatively with albendazole for 12 weeks with a good outcome. The management of spinal hydatid disease is reviewed.     

骨包虫病的诊断及治疗

目的 回顾性分析骨包虫病的临床表现和X线征象，探讨其诊断、鉴别诊断及治疗方法。方法 自1957年10月～2004年2月收治的骨包虫病患者37例，男16例，女21例。年龄14～58岁，平均29岁。病史0．5～12年，平均3．1年。发病部位：颈椎2例，肩胛骨1例，胸椎11例，肋骨2例，腰椎5例，髂骨1例，骶骨6例，耻骨1例，髋关节2例，股骨转子间1例，股骨上段2例，胫骨上段1例，肱骨头1例，桡骨上段1例。所有患者均采用手术治疗，以病灶清除为主，部分患者行自体骨、异体骨、人工骨或骨水泥填塞。术后口服阿苯达唑(片剂或原粉每日20mg／kg；阿苯达唑脂质体每日10mg／kg)3个月，预防复发。结果 25例行皮内Casoni试验，阳性2l例(84%）。4例行包虫病八项免疫试验，均为阳性。21例行MR检查，18例诊断为骨包虫病。24例获随访者，随访时间2～20年，平均3．6年。11例复发(45．83％)，其中胸椎4例，腰椎2例，骶骨4例，髋关节l例。6例四肢骨包虫病患者未见复发。24例脊柱骨包虫病患者中16例有神经系统损害，Frankel分级A级l例，B级3例，C级3例，D级9例，所有病例术后均有不同程度改善。结论 骨包虫病好发于躯干骨，尤其是脊柱。其影像学表现与结核、转移癌、骨巨细胞瘤、骨囊肿等相似，应注意鉴别。MRI有助于脊柱包虫病的诊断，血清学检查是诊断骨包虫病的主要方法；脊柱包虫病伴脊髓压迫采用手术治疗症状改善明显，但病灶不易清除彻底，容易复发。     

Primary Spinal Intradural Extramedullary Hydatid Cyst in a Child

Abstract

Background/Objective: Spinal hydatid cyst is a serious form of hydatid disease affecting less than 1% of the total cases of hydatid disease. We present a case of pathologically confirmed primary intradural spinal cyst hydatid in an otherwise healthy patient who showed no other evidence of systemic hydatid cyst disease.

Case Report: An 8-year-old boy presented with back pain, left leg pain, and difficulty in walking. The patient had no other signs of systemic hydatid cyst disease. An intradural extramedullary cystic lesion was identified with magnetic resonance imaging and was shown to be a hydatid cyst by histopathologic examination after the surgical removal.

Conclusion: Although extremely rare, primary intradural extramedullary hydatid cyst pathology might be the cause of leg pain and gait disturbance in children living in endemic areas.     

评估X、CT及MRI在脊柱包虫病诊断中的价值

目的 回顾性分析25例脊柱包虫病的临床表现和影像特点,探讨其诊断及误诊原因.方法 1957年10月至2006年6月收治5721例包虫病中,脊柱包虫病患者25例,男14例,女11例,年龄15-56 岁,平均28.3岁,病史0.5-12 年,平均3.2年.发病部位:颈椎3例,胸椎11例,腰椎5例,骶骨6例.从临床、流行病学、影象学及免疫学等方面综合分析,总结其影像学特点及误诊原因.结果 25例脊柱包虫中有23例来自农牧区或有犬羊密切接触史(92%).25例均行X线检查,13例行CT检查,5例(38.46%)诊断包虫.19例行MR检查,17例(89.47%)诊断为包虫病,多房性是本病的特征之一,T1WI母囊信号高于子囊是其另一特征.15例行皮内casoni试验,阳性12例(80%),4例行包虫病八项免疫试验均为阳性.结论 脊柱包虫病X线和CT表现易误诊为结核、转移瘤、骨巨细胞瘤或骨囊肿等,MRI有助于脊柱包虫的诊断与鉴别诊断,血清学检查对确诊诊断有很大帮助.     

Dumbbell hydatid cyst of the spine: case report and review of the literature

STUDY DESIGN: A case report of a hydatid cyst in the retropleural space at T7-T8 with an extension into the spinal canal through an intervertebral foramen complicated by paraparesis due to thoracic cord compression. OBJECTIVE: To confirm that hydatid cyst should be considered in the differential diagnosis for any mass discovered in the human body. SUMMARY OF BACKGROUND DATA: Appearance of a dumbbell-shaped mass in the thoracic spine is highly suggestive of neurofibroma. Hydatid cyst, although very rare, may have the same appearance. The spine is involved in 50% of hydatid disease of the bone. A hydatid cyst of the bone and muscle is always primary. Neurologic recovery is possible because it is a slow-growing lesion. METHOD: Clinical symptoms, differential diagnosis, and treatment are reviewed. The patient was treated successfully by total surgical excision of the lesion through a posterior retropleural approach. No medical treatment was prescribed. RESULTS: Surgical excision is effective in the treatment of hydatid disease of the spine, particularly if there is no extensive bony destruction and it is possible to achieve neurologic recovery after decompression of the thoracic spine. CONCLUSION: Extra spinal hydatidosis may lead to spinal cord compression by extension through the intervertebral foramen. The possibility of cure is high when there is no bony involvement.     

Spinal hydatidosis accompanied by a secondary infection. Case report

Spinal hydatid disease is rarely encountered in nonendemic regions. It is a progressive disease that is associated with risks of serious morbidity. The authors report a case of an isolated primary hydatid cyst of the spine in a 34-year-old woman who presented with back pain, which had lasted for 3 months, as well as lower-extremity pain and fatigue, which had persisted for 2 months. A neurological examination yielded findings indicative of upper motor neuron involvement with complete sensory loss below the level of T-11. Magnetic resonance imaging of the spine showed multiple extradural cystic lesions with an abscess formation at T-11 and involvement of the paraspinal muscles. The patient underwent spinal decompression in which stabilization and total excision of the multiple epidural and psoas abscesses and paraspinal multiloculated cysts were performed. The diagnosis of hydatid disease associated with another infection was confirmed by histopathological evaluation. Albendazole was administered during the postoperative period. Previous reports of secondary infections accompanying this disease in extraspinal locations have been published. However, to the best of the authors' knowledge, there have been no publications about a secondary infection associated with hydatidosis in the spinal cord. One should bear in mind that spinal hydatidosis may be accompanied by other infections in endemic regions. Antihelminthic treatment should be administered for a long period following early decompressive surgery and adequate stabilization.     

Disseminated intraspinal hydatid disease

Spinal echinococcosis is a rare entity, accounting for 1% of all cases of hydatid disease. The authors report the case of a 60-year-old man whom they treated for recurrent nerve root compression due to disseminated intraspinal echinococcosis (hydatid disease). Six years previously he had undergone surgery on an emergency basis at another institution after presenting with acute paraplegia due to a primary extradural hydatid cyst of the thoracic spine. Unfortunately, during surgical removal of the cysts, the echinococcosis disseminated into the spinal canal. This complication was documented by magnetic resonance (MR) imaging. In the 4 years before the authors treated him, he was hospitalized 4 times for 4 recurrences of nerve root compression. The authors treated the disseminated disease successfully with total T7-8 corpectomy, grafting with titanium cage and Texas Scottish Rite Hospital instrumentation, and long-term administration of albendazole (400 mg daily). Early diagnosis, proper utilization of MR imaging, and radical resection of diseased vertebrae and soft tissues followed by anthelmintic treatment are essential to control disseminated spinal hydatidosis and prevent recurrence.