CPQ Neurology and Psychology (2019) 1:5
Case Report

Paraplegia Revealing Primary Extradural Spinal Hydatidosis

Salem Bouomrani1,2*, Nizar Khlass1, Moez Ben Ayed2,3, Oussema Souissi1 & Nesrine Regaïeg1,2

1Department of Internal Medicine, Military Hospital of Gabes, Tunisia
2Sfax Faculty of Medicine, University of Sfax, Tunisia
3Department of Orthopedics, Regional Hospital of Gabes, Tunisia

*Correspondence to: Dr. Salem Bouomrani, Department of Internal Medicine, Military Hospital of Gabes, Tunisia.

Copyright © 2019 Dr. Salem Bouomrani, et al. This is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

Received: 18 April 2019
Published: 17 May 2019

Keywords: Paraplegia; Extradural Hydatid Cyst; Spine; Hydatidosis; Echinococcosis


Spinal hydatidosis is exceptional: prevalence estimated at less than 1% of all localizations of the hydatid disease. Extradural forms of spinal hydatid cyst are rare and only a few sporadic cases have been reported in the medical literature. It is characterized by its high clinical latency, potential severity, and frequent recurrence. It can present itself as a real neurological emergency: spinal cord compression and cauda equina syndrome. It is thus recommended to evoke this diagnosis in front of any symptomatology of non-traumatic spinal cord compression in endemic areas for this parasitosis.

We report an original observation of acute paraplegia revealing a primary extradural hydatid cyst of the dorsal cord in a young Tunisian man.

Echinococcosis is an anthropozoonosis that is still epidemic in several countries of the world, particularly in rural areas where there is an intimate contact between humans and animals (mainly sheep and dogs) [1].

The main localizations of this parasitosis are the liver and the lungs: respectively 65-70% and 25% of the cases [1,2].

More rarely, other unusual locations [3] described as “ectopic” and “aberrant” [4] can be noted and represent real diagnostic challenges for clinicians [5-10].

Hydatid disease of the central nervous system (CNS) remains rare compared to other unusual locations; it accounts for only about 2% of all human echinococcosis [11]. It affects the brain in 2/3 of the cases and the marrow in 1/3 of cases [12]. The prevalence of spinal hydatidosis is thus estimated at less than 1% of all localizations of hydatid disease [13]. Extradural forms of spinal hydatid cyst (HC) are rare and only a few sporadic cases have been reported in the medical literature.

We report an original observation of acute paraplegia revealing a primary extradural HC of the dorsal cord in a young Tunisian man.

Case Report
A 23-year-old man, with no particular pathological history, presented to the emergency department for lower limb weakness evolving for 3 months with progressive installation and worsening. The walk became impossible the morning of his consultation at the emergency room. The examination showed paraplegia with hyperreflexia of the lower limbs, and a sensory level in T4. The patient was apyretic, eupnetic, conscious, oriented, and with stable hemodynamic state and vital constants.

Basic biological tests (total blood count, postprandial glucose, C-reactive protein, erythrocyte sedimentation rate, ionogram, calcium, magnesium, muscle enzymes, lipid parameters, transaminases, thyroid hormones, and electrophoresis of serum proteins), the electrocardiogram, and the chest x-ray were without significant abnormalities.

Medullary magnetic resonance imaging (MRI) revealed a cystic, extradural, multi-vesicular lesion, extended from T5 to T8, and compressing the medullary cord. The lesion was hypointense on T1-weighted images, hyperintense on T2-weighted images, and with no enhancement after gadolinium injection (Figs 1, 2 and 3). Thoraco-abdominopelvic CT did not note other associated visceral lesions.

Figure 1: T2-weighted sagittal spine MRI: extradural, multi-vesicular cystic mass, extending from T5 to T8, and with spontaneous hypersignal

Figure 2: T2-weighted axial spinal MRI: extradural, multi-vesicular cystic lesion, and with spontaneous hypersignal

Figure 3: T1-weighted sagittal spine MRI without (a) and with (b) gadolinium injection: extradural, multivesicular mass, extending from T5 to T8, hypointense, and without secondary enhancement

A T5-T8 laminectomy was performed showing an extradural cyst with a colorless content suggestive of a HC. The cyst was removed completely (total cystectomy).

Histopathological examination confirmed the diagnosis of extradural spinal HC. The hydatid serology was negative and subsequent investigations did not reveal other hydatid locations confirming the primary character of the spinal HC. Postoperatively, the patient received oral albendazole at a dose of 800mg/day for 4 months.

Progressive improvement was noted with resumption of walking and muscle strength. The neurological examination was strictly normal after six months of surgery. No recurrence was noted during a two-year postoperative follow-up.

The thoracic spine represents the preferred seat of spinal hydatidosis (52%), followed by the lumbar spinal (37%), then cervical and sacral levels [14,15].

Clinically, the majority of patients presented with back pain and/or lower limb weakness. More rarely sphincter disorders or even a cauda equina syndrome [11-15]. Apart from this symptomatology due to spinal cord compression, spinal hydatid disease has no specific manifestations [11-14].

In endemic countries, spinal HC is among the medical pathologies frequently responsible for medullary compression: 14% of spinal cord compressions were of hydatic origin in Bettaieb A et al series [16]. It is therefore recommended to discuss this diagnosis in front of symptoms of non-traumatic spinal cord compression in endemic areas for echinococcosis [16].

Biologically, eosinophilia is found only in less than 15% of cases and is particularly important in cases of cyst rupture [11-14]. The hydatid serology is also not very contributive to the diagnosis of the spinal localization of HC; it is only positive in about 25% of cases [17]. It is particularly useful for post-operative surveillance to detect recurrences [12,18,19].

MRI is the exam of choice for the positive diagnosis of spinal hydatidosis [20,21], and the radiological classification proposed by Braithwaite and Lees in 1981 distinguishes 5 types of spinal HC: Primary cyst in the cord, Intradural cyst, Extradural cyst, Hydatid disease of the vertebrae, and Paravertebral hydatid disease [22].

The first three types are considered rare [19]; the primary intramedullary type remains exceptional with only a few sporadic cases reported in the literature [13,22-24].

The treatment of choice for spinal HC is surgical [25]. Adjuvant chemotherapy with Albendazol perioperatively is classically recommended to inactivate the cyst preoperatively, and reduce postoperative recurrence [26]. Recurrences are frequent for this localization: 30 to 100% of the cases according to the series [14], and this could be explained by the difficulty of complete excision of the cyst and its sometimes unavoidable rupture, given the narrowness of the operative field and the close relationships with adjacent structures [14,25,26].

The primitive spinal localization of human echinococcosis remains exceptional and unusual. It represents a real diagnostic challenge for clinicians given its serious neurological complications and often urgent presentations.

A better knowledge of this location is the only guarantee of appropriate and urgent care to improve the prognosis often reserved.

It is thus recommended to evoke the diagnosis of spinal HC in front of any spinal symptomatology or spinal cord compression that remains unexplained in endemic areas for this parasitosis.

Conflict of Interest: None


  1. Grosso, G., Gruttadauria, S., Biondi, A., Marventano, S. & Mistretta, A. (2012). Worldwide epidemiology of liver hydatidosis including the Mediterranean area. World Journal of Gastroenterology, 18(13), 1425-1437.
  2. Moro, P. & Schantz, P. M. (2009). Echinococcosis: a review. International Journal of Infectious Diseases, 13(2), 125-133.
  3. Gun, E., Etit, D., Buyuktalanci, D. O. & Cakalagaoglu, F. (2017). Unusual locations of hydatid disease: A 10-year experience from a tertiary reference center in Western Turkey. Ann Diagn Pathol., 29, 37-40.
  4. Saadi, A., Bouzouita, A., Cherif, M., Rebai, M. H., Kerkeni, W., Ayed, H., et al. (2015). Retrovesical hydatic cyst: About 4 cases. Can Urol Assoc J., 9(5-6), E374-E378.
  5. Keser, S. H., Selek, A., Ece, D., Barişik, C. C., Şensu, S., Geçmen, G. G., et al. (2017). Review of Hydatid Cyst with Focus on Cases with Unusual Locations. Turk Patoloji Derg., 33(1), 30-36.
  6. Bouomrani, S., Souissi, O., Guermazi, M., Yahyaoui, S. & Trabelsi, S. (2019). An Unexpected Etiology of Myocardial Ischemia in Young. Journal Of Hypertension And Cardiology, 2(4), 13-19.
  7. Bouomrani, S. & Feki, W. (2019). Pancreatic and gastrointestinal hydatidosis. Gastroenterol Hepatol Open Access, 10(2), 108-110.
  8. Bouomrani, S. & Feki, W. (2019). Humain Splenic Hydatidosis: A Review. Gastroenterol Hepatol Int J., 4(1), 1-6.
  9. Bouomrani, S., Souissi, O., Regaïeg, N. & BenHamed, M. (2018). Primary Hydatid Cyst of the Thyroid Gland Revealed by Primary Hypothyroidism. J Inf Dis Trav Med., 2(3), 000121.
  10. Bouomrani, S., Dabboussi, S., Nefoussi, M., Trabelsi, S. & Baïli, H. (2018). Juvenile Primary Mediastinal Hydatid Cyst. EC Pulmonology and Respiratory Medicine, 7(11), 1-5.
  11. Tizniti, S., Allali, N., El Quessar, A., Chakir, N., El Hassani, M. R. & Jiddane, M. (2000). An unusual cerebral hydatid cyst. J Neuroradiol J Neuroradiol., 27(3), 200‑202.
  12. Limaiem, F., Bellil, S., Bellil, K., Chelly, I., Mekni, A., Khaldi, M., et al. (2010). Primary hydatidosis of the central nervous system: a retrospective study of 39 Tunisian cases. Clin Neurol Neurosurg., 112(1), 23‑28.
  13. Zhang, Z., Fan, J., Dang, Y., Xu, R. & Shen, C. (2017). Primary intramedullary hydatid cyst: a case report and literature review. Eur Spine J., 26(Suppl 1), 107-110.
  14. Schnepper, G. D. & Johnson, W. D. (2004). Recurrent spinal hydatidosis in North America. Case report and review of the literature. Neurosurg Focus., 17(6), E8.
  15. Adilay, U., Tuğcu, B., Gunes, M., Günaldi, O., Gunal, M. & Eseoglu, M. (2007). Cauda equina syndrome caused by primary lumbosacral and pelvic hydatid cyst: a case report. Minim Invasive Neurosurg, 50(5), 292-295.
  16. Bettaieb, A., Khaldi, M., Ben Rhouma, T. & Touibi, S. (1978). Spinal echinococcosis; clinical study of 32 cases. Neurochirurgie, 24(4), 205-210.
  17. Kotil, K., Tatar, Z. & Bilge, T. (2007). Spinal hydatidosis accompanied by a secondary infection. Case report. J Neurosurg Spine., 6(6), 585-590.
  18. Turgut, M. (1997). Hydatid disease of the spine: a survey study from Turkey. Infection, 25(4), 221-226.
  19. Lotfinia, I., Sayyahmelli, S., Mahdkhah, A. & Shoja, M. M. (2013). Intradural extramedullary primary hydatid cyst of the spine: a case report and review of literature. Eur Spine J., 22(Suppl 3), S32936.
  20. Berk, C., Ciftçi, E. & Erdoğan, A. (1998). MRI in primary intraspinal extradural hydatid disease: case report. Neuroradiology, 40(6), 390-392.
  21. Tsitouridis, I. & Dimitriadis, A. S. (1997). CT and MRI in vertebral hydatid disease. Eur Radiol., 7(8), 1207-1210.
  22. Braithwaite, P. A. & Lees, R. F. (1981). Vertebral hydatid disease: radiological assessment. Radiology, 140(3), 763-766.
  23. Ley Jr, A. & Marti, A. (1970). Intramedullary hydatid cyst. Case report. J Neurosurg., 33(4), 457-459.
  24. Senol, M. G., Tekeli, H., Kendirli, M. T., Kaya, S., Turhan, V., Sonmez, G., et al. (2012). Intramedullary hydatid cyst of the cervical spine. Indian J Med Microbiol., 30(4), 480-481.
  25. Gennari, A., Almairac, F., Litrico, S., Albert, C., Marty, P. & Paquis, P. (2016). Spinal cord compression due to a primary vertebral hydatid disease: A rare case report in metropolitan France and a literature review. Neurochirurgie, 62(4), 226-228.
  26. Salduz, A., Koyuncu, L. O., Dikici, F. & Talu, U. (2009). Long-term result of treatment for paraspinal and extradural hydatid cyst: a case report. Acta Orthop Traumatol Turc., 43(3), 267-271.

Total Articles Published


Total Citations:



Cient Periodique is a ‘Gold’ open access publisher that aspires to offer absolute free, unrestricted access to the valuable research information

We welcome all the eminent authors to submit your valuable paper

Cient Periodique invites the participation of honourable Editors and Authors

CPQ Journals provide Certificates for publication

Cient Periodique also offers memberships for potential Authors

Best Articles will be appreciated with the provision of corresponding Certificate


We're here to answer your questions!

Send us a message via Whatsapp, and we'll reply the moment we're available!