Paraplegia Associated With Spinal Hydatid Cyst

SPINE Volume 35, Number 9, pp E356 –E358 ©2010, Lippincott Williams & Wilkins Paraplegia Associated With Spinal Hydatid Cyst A Case Report Canan Celik, MD,* Munevver Fatma Sasmaz, MD,* Fugen Oktay, MD,* Halil Ucan, MD,* and Erkan Kaptanoglu, MD† Study Design. Case report. Objectives. To report a case with paraplegia caused by spinal hydatid cyst. Summary of Background Data. Hydatid cyst is a disease caused by larval Echinococcus granulosus tapeworm. Spinal hydatid cyst rarely leads to severe neurologic problems. Methods. A 34-year-old male patient was referred to our outpatient clinic due to back and low back pain, progressive weakness and numbness in both lower extremities, and a prediagnosis of lumbar disc hernia. He had spastic paraplegia, and thorax magnetic resonance imaging revealed a lobulated cystic lesion with extradural intraspinal localization. Results. After surgery and following 2 months of rehabilitation program, the patient showed a dramatic clinical improvement. Conclusion. By this case, it is emphasized that spinal hydatid cyst should come to mind in the differential diagnosis of spinal cord compression, and the importance of prevention, early diagnosis, and treatment is highlighted because of high mortality and morbidity. Key words: Echinococcus, hydatid cyst, spinal cord compression. Spine 2010;35:E356 –E358 Hydatid cyst is a disease caused by the larval Echinococcus granulosus tapeworm.1 The disease is spread by animals, thus, it is seen in all countries, especially in the Mediterranean countries, Turkey, Africa, Middle Eastern and Asian countries, East and South Europe, Latin and South America, Australia, and New Zealand. E. granulosus mostly involves liver (50%– 60%) and lungs (20%–30%), however, any organ of the body can also be involved (⬍10%).2 Only 0.5% to 3.1% of patients have bone involvement and half of them have concomitant spinal involvement.3 Case Report We evaluated a 34-year-old male patient, who was a farmer, presented with back and low back pain, progresFrom the *Department of Physical Medicine and Rehabilitation, Ankara Physical Medicine and Rehabilitation Education and Research Hospital; and †Department of Neurosurgery, Ankara Numune Education and Research Hospital, Ankara, Turkey. Acknowledgment date: May 5, 2009. First revision date: August 10, 2009. Acceptance date: October 7, 2009. The manuscript submitted does not contain information about medical device(s)/drug(s). No funds were received in support of this work. No benefits in any form have been or will be received from a commercial party related directly or indirectly to the subject of this manuscript. Address correspondence and reprint requests to Canan Celik, MD, Tirebolu Sokak No: 53/10, 06550 Y, Ayranci/Ankara, Turkey; E-mail: ccelik@hotmail.com E356 sive weakness and numbness in lower extremities, which had been observed 2 months previously. His prediagnosis was lumbar disc hernia, and he was consulted to our clinic for further evaluation. His past medical history was remarkable for hydatid cyst in the liver, and he had a liver surgery 4 years ago. On physical examination, the patient was no ambulatory. Neurologic examination revealed that lumbar spine movements were painful and range of motions was limited in all ranges. There was also paravertebral spasm in this region. Muscle strength of the left and right lower extremities was 2/5 and 3/5, respectively. Bilateral lower extremities are mildly spastic (I in Ashworth scale), patella and Achilles reflexes were hyperactive, and plantar response was bilaterally extensor. Superficial abdominal cutaneous reflex was absent. Touch and pain sensation were normal until T7, and there was patchy hypoesthesia below this level. Bladder and bowel functions were preserved, but he had frequent urination. Anal reflex, anal tonus, and anal contraction were present. Complete blood cell count and blood biochemistry were within the normal limits. Thorax roentgenograms were normal. Lumbar magnetic resonance imaging (MRI) of the patient showed multilocational degenerative changes. The thoracic MRI demonstrated a lobulated cystic lesion with extradural expanding and invading the left lamina, pedicle, and transverse processes of T5-T6 vertebrae, extending to the costae, forming a “scalloping” effect on T5 and T6 vertebrae via the posterior of the left corpus, extending to the spinal canal, and compressing the spinal cord to right and anterior, showing hypointensity at T1W- and hyperintensity at T2Wimages while showing no contrast uptake following intravenous contrast material injection, extending to the left paravertebral extrapleural region (Figures 1–2). The spinal cyst localized extradurally and intraspinally with vertebral, paravertebral, and multicompartment involvement. The Echinococcus serology that was performed with the prediagnosis of hydatid cyst revealed positive indirect hemagglutination test at 1/160 titer, and the eosinophil count was 3%. Emerge surgical procedure with posterior approach was performed to extract the spinal cyst. Laminectomy and costotransversectomy were performed at T6 level, along with the excision of extradural cysts. The pathology report was compatible with hydatid cyst. Cyclic treatment with albendazole 800 mg/d was started. After the surgery, the pa- Paraplegia Associated With Spinal Hydatid Cyst • Celik et al E357 Figure 1. T2W axial images showing a lobulated contoured cystic lesion (hydatid cyst) invading the left lamina, pedicle, and transverse process of T6 vertebrae, extending to the spinal canal and compressing the spinal cord to the right and anterior. tient was transferred to the rehabilitation clinic. After the rehabilitation program, muscle strength of both lower extremities had increased to 4/5. Sensory, tonus, and deep tendon flexes were normal, while plantar response was flexor. Superficial abdominal cutaneous reflex was present. Anal reflex, anal tonus, and voluntary anal contraction were present. In urodynamic analysis, hyperreflex synergic bladder was detected, and anticholinergic treatment was started. Urination frequency decreased. The patient was discharged in independent ambulatory status. Discussion Spinal hydatid disease is an infectious disease characterized by multivesicular diffuse infiltration of larvae of E. granulosus to cancellous bone, including corpus vertebrae, pedicles, and the lamina.3 The present case revealed the intraspinal extradural location with multicompartment involvement of hydatid cyst. There are no characteristic signs and symptoms for spinal hydatid cyst. In a meta-analysis performed in Turkey, the rates of common signs were as follows: weakness in lower extremities 73%, low back pain 43%, bladder/intestine dysfunction 32%, pain in lower extremities 27%, sensation disorders 24%, and paravertebral swelling 2%.4 In our case, the first symptoms were back and low back pain and paraplegia. Diagnosis of such cases is difficult and frequently delayed until signs and symptoms of spinal cord and nerve compression develop.1,5 MRI is the most sensitive diagnostic method. Fahl et al6 stated that cysts usually look like flattened sausages and have 2 dome-shaped ends, consisting of thin and regular walls without septums or debris within the lumen. Lesions are sometimes spherical. Signal characteristics of the cyst content are usually similar to that of cerebrospinal fluid. In the present case, Figure 2. T2W sagittal images showing extradural spinal cyst, which is a sausage-like shape with 2 dome shaped ends in dorsal spine. the MRI images and signal characteristics were in accordance with these definitions. Vertebral tuberculosis, pyogenic infections, fibrous dysplasia, enchondroma, malignancies, multiple myeloma, giant cell tumors, and hyperparathyroidism should come to mind in the differential diagnosis.1 Misdiagnosis can frequently be the case.1,4 In the treatment of spinal hydatid cyst, surgery is still accepted as the “gold standard.”1,3 Because of high recurrence rates, it has a poor prognosis and has been compared with spinal malignancy. 1,4 In the present case, previous liver operation for hydatid cyst was conspicuous. Conclusion Spinal hydatid cyst can also be seen in nonendemic countries because of traveling of people all over the world. Physicians should be careful in the differential diagnosis of patients with spinal pain, radicular pain, progressive myelopathy, or spinal cord compression syndrome. Be- E358 Spine • Volume 35 • Number 9 • 2010 cause of high mortality and morbidity; importance of prevention, early diagnosis, and treatment is highlighted. Key Points ● ● ● ● Spinal hydatid cyst rarely leads to severe neurologic problems and paraplegia. It was evaluated a patient whose the first symptoms were back and low back pain and paraplegia. A dramatic clinical improvement has been observed after surgery and rehabilitation program. This disease should come to mind in the differential diagnosis of spinal cord compression. References 1. Schnepper GC, Johnson WD. Recurrent spinal hydatidosis in North America. Case report and review of the literature. Neurosurg Focus 2004; 17:E8. 2. King CH. Cestodes. In: Mandell GL, Bennett JE, Dolin R, eds. Mandell, Douglas and Bennett’s Principles and Practice of Infectious Diseases. 6th ed. Philadelphia: Elsevier Churchill Livingstone Inc.; 2005; 3285–93. 3. İçslekel S, Erçsahin Y, Zileli M, et al. Spinal hydatid disease. Spinal Cord 1998;36:166 –70. 4. Turgut M. Hydatid disease of the spine: a survey study from Turkey. Infection 1997;25:221– 6. 5. Fahl M, Haddad FS, Huballah M, et al. Magnetic resonance imaging in intradural and extradural spinal echinococcosis. Clin Imaging 1994;18: 179 – 83. 6. Rkain H, Bahiri R, Benbouaza K, et al. An extensive vertebral hydatidosis revealed by a lumbosciatica. Clin Rheumatol 2007;26:1386 – 8.