|Year : 2012 | Volume
| Issue : 4 | Page : 480-481
Intramedullary hydatid cyst of the cervical spine
Senol, Mehmet Güney, Tekeli, Hakan Kendirli, Mustafa Tansel, Kaya, Serdar, Turhan Vedat, Sonmez Güner, Saracoglu, Meh
GATA, Haydarpasa Training Hospital, Istanbul, Turkey
|Date of Submission||15-Apr-2012|
|Date of Acceptance||22-Jul-2012|
|Date of Web Publication||24-Nov-2012|
GATA, Haydarpasa Training Hospital, Istanbul
Source of Support: None, Conflict of Interest: None
Hydatid disease (Echinococcosis) is a common parasitic infection caused by Echinococcus granulosus mainly in sheep-raising areas of the world. Liver, lungs and brain are the predominantly involved organs. However, 0.5-1% of the hydatid disease involves the spine and in 90% of the cases it is confined to the bone and the epidural space. Although intramedullary involvement is extremely rare, in this report, we present a 55-year-old female patient who was diagnosed with a cervical intramedullary hydatid cyst during magnetic resonance imaging of the cervical vertebrae. Accordingly, we imply that particularly in endemic areas, hydatid cyst disease should be kept in mind for the differential diagnosis of spinal mass lesions.
Keywords: Cervical intramedullary cyst, Hydatid disease, magnetic resonance imaging
|How to cite this article:|
Senol, Güney M, Tekeli, Kendirli H, Tansel M, Kaya, Serdar, Vedat T, Güner S, Saracoglu, Meh. Intramedullary hydatid cyst of the cervical spine. Indian J Med Microbiol 2012;30:480-1
|How to cite this URL:|
Senol, Güney M, Tekeli, Kendirli H, Tansel M, Kaya, Serdar, Vedat T, Güner S, Saracoglu, Meh. Intramedullary hydatid cyst of the cervical spine. Indian J Med Microbiol [serial online] 2012 [cited 2019 Aug 22];30:480-1. Available from: http://www.ijmm.org/text.asp?2012/30/4/480/103780
| ~ Introduction|| |
Hydatid disease is caused by the larvae of the tapeworm Echinococcus granulosus. Echinococcus multilocularis may also cause hydatid disease. It is endemic in Mediterranean, Eastern European, East African, Central Asian and South American countries, Australia and New Zealand.  While the most commonly involved tissues include liver (50-70%), lungs (20-30%) and brain (2-4%); the disease may ensue in the heart, bone or other organs of the body as well. ,
Spinal involvement has been reported in only 0.5-1% of the cases where 90% of them are confined to the vertebrae and the epidural space.  Spinal hydatid disease has been classified by Braithwaite and Lees into five types: (1) primary intramedullary hydatid cyst, (2) intradural extramedullary hydatid cyst, (3) extradural intraspinal hydatid cyst, (4) hydatid disease of the vertebrae and (5) paravertebral hydatid disease.  The first three types are rarely reported.  To the best of our knowledge, only one case of intramedullary hydatid cyst of the spine has been reported in the medical literature.  In this report, the second case of intramedullary hydatid cyst in the cervical spine is presented.
| ~ Case Report|| |
A 55-year-old female patient was admitted to our hospital with complaints of headache, pain on her left arm and numbness on the left side of her body for the last 3 months. Her past medical history includes autoimmune thyroid disease and also migraine headache for more than 20 years . In addition she grew up in a rural area and was in frequent contact with farm animals. Increased deep tendon reflexes in the upper extremities were the only positive finding in her neurological examination. Since her cranial magnetic resonance imaging (MRI) demonstrated a suspicious lesion, cervical MRI was also performed and it revealed a non-contrast enhancing cystic lesion (1.2 × 0.7 cm) which was hypointense on T1-weighted and hyperintense on T2-weighted images [Figure 1]. Thoracic and abdominal computed tomography (CT) showed multiple cysts in her liver, spleen and lungs. A paraspinal cystic lesion (2.5 × 1 cm) at the level of 5 th and 6 th thoracic vertebrae was also documented [Figure 1]. Radiological appearances of the cysts were consistent with hydatid disease. Serological tests including enzyme linked immunosorbent assay (ELISA), indirect hemagglutination (IHA) and immunofluroscent detection of antibodies (IFA) against Echinococcus granulosus confirmed the diagnosis of hydatid disease.
|Figure 1: T2-weighted non-contrast enhancing (-gadolinium) cervical MRI (B) sagittal (D) axial views showing a hyperintense intramedullary mass at the level of C2 vertebra. T1-weighted spinecho image (+gadolinium) (A) sagittal (C) axial views shows cystic hypointense lesion at the level of C2 vertebra. There is a paraspinal cystic lesion at the T5-6 vertebrae (white arrow) (A, B)|
Click here to view
After three courses of albendazole treatment she was scheduled for regular follow-up visits. Cysts in her lung, spleen and liver were successfully removed by consecutive operations. However, because of its high risks, neurosurgeons avoided surgery for intramedullary cyst and decided to follow the patient once a year regularly.
Until this time, the patient was treated several times with a daily dose of 800 mg albendazole, bid, for 28-day cycle followed by 14-day drug-free intervals. There was no remarkable change in size on cervical spinal lesion by contrast-enhanced MRI during the 6 years of follow-up period. However, her clinical symptoms such as pain on her left arm and numbness on the left side of her body gradually got better.
| ~ Discussion|| |
Hydatid disease rarely involves spine.  A number of cases with intradural extramedullary hydatid cysts have been reported.  However, only one case of intramedullary hydatid cyst has been reported in the current literature.  In this report, we presented the second case of intramedullary hydatid cyst of cervical spine.
Although the diagnosis of hydatid disease is mainly established by serological tests, imaging findings provide important hints for the clinicians. CT and MRI findings include the presence of well-defined, smooth thin-walled, homogeneous cysts where the fluid appears to be similar to the cerebrospinal fluid. On unenhanced CT, the cyst wall appears iso- or hyperdense to neural tissue. The cyst wall displays a low signal intensity rim on both T1- and T2-weighted MRI images. However, the cyst is conspicuous on T2-weighted images because of its high signal intensity fluid content. 
En bloc removal surgery is the treatment of choice in hydatid cyst disease. Careful puncture and aspiration of cyst contents, followed by total extirpation of the cyst wall, can be performed when total removal is difficult and hazardous.  Chemotherapy usually includes well known anti-helmintic drugs: albendazole or mebendazole. Gamma knife surgery is suggested for poor surgical candidates with spinal cyst hydatidosis.  Due to benign clinical presentation, multiplicity of the cysts in liver and lungs, and the intramedullary localisation of the spinal cyst, surgery was not performed in our case and the patient was followed with albendazole treatment.
Differential diagnosis of intramedullary cystic masses includes syrinx cavities, myelomalacia, hematoma, arachnoid cyst, neurenteric cyst, epidermoid cyst, cystic hemangioblastoma and astrocytoma like cystic neoplasms.  Our patient's multiple cysts in liver and lungs suggested her intramedullary lesion to be most likely a hydatid cyst.
In conclusion, presenting this interesting case, we imply that particularly in endemic areas, hydatid cyst disease should be kept in mind for the differential diagnosis of spinal cystic lesions. In this regard, MRI is of value for differential diagnosis of spinal cystic lesions.
| ~ References|| |
Bayar MA, Erdem Y, Habip N. Primary intraspinal extradural hydatid disease causing radicular compression. Turk Neurosurg 1997;7:33-5.
Sridhar K, Ramamurthi B. Granulomatous fungal and parasitic infections of the spine. In: Menezes AH, Sonntag VK, editors. Principles of Spinal Surgery. New York: McGraw-Hill; 1996. p. 1467-97.
Delibaº SB, Ozkoç S, Sahin S, Aksoy U, Akisü C. [Evaluation of patients presenting with a suspicion of cystic echinococcosis to the serologylaboratory of the Parasitology Department of Dokuz Eylül University Medical Faculty]. Turk Parazitol Derg 2006;30:279-81.
Charles RW, Govender S, Naidoo KS. Echinococcal infection of the spine with neural involvement. Spine (Phila Pa 1976) 1988;13:47-9.
Braithwaite PA, Lees RF. Vertebral hydatid disease: Radiological assessment. Radiology 1981;140:763-6.
Ley A Jr, Marti A. Intramedullary hydatid cyst. Case Report. J Neurosurg 1970;33:457-9.
Arif SH, Zaheer S. Intradural extramedullary primary hydatid cyst of the spine in a child: A very rare presentation. Eur Spine J 2009;18:179-82.
Tüzün M, Hekimoðlu B. Hydatid disease of the CNS: Imaging Features. AJR Am J Roentgenol 1998;171:1497-500.
Altinörs N, Bavbek M, Caner HH, Erdogan B. Central nervous system hydatidosis in Turkey: a cooperative study and literature survey analysis of 458 cases. J Neurosurg 2000;93:1-8.
Guzel A, Tatli M, Yilmaz F, Bavbek M. Unusual presentation of cervical spinal intramedullary arachnoid cyst in childhood: Case report and review of the literature. Pediatr Neurosurg 2007;43:50-3.
|This article has been cited by|
||Cause rare de lombosciatalgie symptomatique : hydatidose pelvienne
| ||Khaled Bouzaïdi,Alifa Daghfous,Mouna Chelli Bouaziz,Hana Sahli,Lamia Rezgui Marhoul,Mohamed Fethi Ladeb |
| ||La Presse Médicale. 2015; |
|[Pubmed] | [DOI]|