Spinal hydatid cysts

Surg Neurol 1984;21:53-7

53

Spinal Hydatid Cysts M. Necmettin

Pamir, M.D., Nejat Akalan, M.D., Tungalp Ozgen, M.D., and

Aykut Erbengi, M.D. The Department of Neurosurgery, Hacettepe University School of Medicine, Ankara, Turkey

Pamir MN, Akalan N, Ozgen T, Erbengi A. Spinal hydatid cyst. Surg Neurol 1984;21:53-7.

Eleven cases of spinal hydatid cyst are reported. The patients were admitted with symptoms of compression of the spinal cord. The cysts were localized epidurally in 10 patients and intradurally but extramedullarily in one, and were confirmed histopatholgically after surgical intervention. The results are compared with those reported in the literature. Six of the patients were female and five were male. Their ages ranged from 10 to 65 years. All of the patients were treated surgically, with an incidence of cyst recurrence of 18% and no mortality. Mebendazole (Vermox) was given to two of the patients after their operations. KEY WORDS:

Spinal cord compression; Echinococcus; Hydatid

cyst

Hydatid cyst, a lesion that appears during the evolution of the parasite Echinococcusgranulosus, can be found in various sites in the human body, and is localized to the bone in 0 . 5 % - 2 % o f all patients [3,4,6,8,14,16]. Half of this involvement is found in the vertebrae [3,4, 7,8,14,19]. Hydatid cysts are therefore one cause of spinal-cord compression [7]. The purpose of this paper is to describe the incidence, clinical and laboratory findings, treatment, and postoperative results in 11 cases of spinal hydatid cyst disease treated at our center, and to review the literature on this subject.

Material During the past 10 years (1972-1982), 11 patients with spinal hydatid-cyst disease were admitted to the Neurosurgery Department o f Hacettepe University Hos-

Address reprint requests to: Dr. Nejat Akalan, Hacettepe 0niversitesi Tip Fak/~ltesi, Beyin ve Sinir Cerrahisi B61umii, Ankara, Tiirkiye.

© 1984 by Elsevier Science Publishing Co., Inc.

pitals with symptoms of spinal-cord compression requiting surgical intervention. The records, neuroradiologic examinations, surgical findings, and histopathology in these cases are reviewed and the results are described below.

Results The oldest patient was 65 years old and the youngest 10. O f the patients, 54% were under 30 years old. Six of the patients were female (54.6%) and five were male (45.4%). The major complaints were loss of ability to move the legs (six patients), loss of strength in the legs (two patients), and back pain radiating to the legs (three patients) (Table 1). The time interval between the beginning of these complaints and admission to the hospital averaged 3.5 weeks. It was learned that four of the patients had had previous surgical intervention for their condition, with the diagnosis of "hydatid cyst." Upon neurological examination, five of the patients were paraplegic and six were paraparetic. In every case, other neurological sysmptoms gave evidence of compression of the spinal cord (Table 1). Plain radiologic examinations of the patients revealed a pulmonary hydatid cyst in one and compression fractures of vertebral bodies in two others (Table 2); in one of these (case 7) the compression was at Th-9 and in the other (case 10) at L-3. In case 10, plain radiograms also revealed the calcification of tissues adjacent to the compressed vertebral body. Routine x-ray studies and tomographic examinations o f the lumbar vertebrae in case 11 revealed erosion of the dorsal part of the L-5 vertebral body and sacrum. Myelography was done on all o f the patients, with the complete blockage of flow of the contrast medium, indicating epidural or intradural compression, being found in all (Table 2). All of the patients underwent surgical intervention. In 10 the operative technique was a laminectomy, and in 1 a transthoracic vertebrectomy and anterior fusion. In 2 of the 10 patients cysts were seen in the paraver0090-3019/84/$3.00

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Surg Neurol 1984;21:53-7

Pamir et al

Table 1. Age and Sex Distribution, Initial Complaints, and Neurological Findings of these Patients with Spinal Hydatid Cysts Case no.

Age

Sex

Complaints

Duration

1

19

M

Low back pain Impairment o f leg movements Back pain Impairment of leg movements Impairment of leg movements

1.5 mo

2

3

4

5

45

65

44

40

F

F

M

F

6

30

F

7

25

F

8

10

M

9

34

10

11

Previous surgical procedure Cyst excision from back 8 m o earlier

2 mo

3 mo

Impairment of leg movements

40 days

Impairment of leg movements

1 wk

Weakness at leg movements Pain at legs Pain at right leg

1 yr

Hydatid cyst; excision by thoracotomy 18 yr earlier Cyst excision from back 1.5 yr earlier

5 mo

Surgical intervention for Pott's disease

Impairment of leg movements

15 days

Excision of thoracic hydatid cyst 2.5 yr earlier

F

Back pain weakness at legs

l yr

29

M

Back pain

3 yr

12

M

Back pain

1 mo

Neurological findings Paraparesia Sensory loss Th-8 Paraplegia Sensory loss Th-8 Paraplegia Sensory loss Th-5 Paraplegia Sensory loss Th-5 Paraplegia Sensory loss Th-5 Paraplesia

below

below

below

below

below

Paraparesia Sensory loss below Th-10 Paraplegia Sensory loss below Th-10 Paraparesia Sensory loss below Th-10 Global weakness at left leg Sensory loss below L-1 Paraparesia Sensory loss below L-1

Abbreviations: wk = week; mo = month; yr = year.

tebral muscles beneath the muscle fascia. In 10 cases the cysts were located epidurally, while in one the cyst was intradural but extramedullary (Table 3). The number of cysts excised ranged from one to 70 in each case. In two cases (cases 10 and 11), the epidurally located cysts extended into the vertebral bodies, and an anterior approach was therefore planned for their decompression. From the disk space, multiple cysts were excised, and anterior fusions were later made with grafts prepared from one of the ribs. In every case the surgically treated areas were washed with hypertonic saline solution. In three of the five preoperatively paraplegic patients, movements of the lower extremity were regained in the early postoperative period. In one patient who was paraplegic preoperatively, no remarkable neurological recovery was seen, while in one other patient, only control of the bladder was regained. The neurological deficits receded in four of the six patients with paraparesis, but there was no remarkable change in the other two (Table 3).

Four patients did not return after being discharged. The patients in cases 7, 10, and 11 were found to be normal at postoperative examination. The patient in case 4, who was paraparetic before operation, was able to urinate spontaneously a year later. The patient in case 2, who was paraplegic before operation, was definitely normal 1.5 months postoperatively, but had a thoracotomy for a pulmonary hydatid cyst 5 years after his first operation and returned with paraparesis in the eighth postoperative year. This patient was examined and reoperated upon, and multiple cysts were excised from the same level as in his first operation. The patient in case 3, who was paraplegic before operation, was normal for 3 years thereafter. Reoperation was later done because of a recurring paraparesis, and multiple cysts were excised from the same level as in the first operation. This patient was found to be normal at the end of the fifth year after the second operation (Table 3). In two patients (cases 10 and 11), mebendazole (Vermox-Sonsen) was used postoperatively.

Spinal Hydatid Cysts

Surg Neurol 1984;21:53-7

Table 2. Plain Radiologic Examinations and Myelographic Findings Case no.

Plain Radiograms

1

Normal

2 3

Normal Normal

4

Normal

5

Appearance resembling hydatid cyst in chest radiogram Normal

6 7 8

Compression fracture at Th-9 Normal

9

Normal

10 11

Compression fracture at L-3 Lyric lesion at sacrum

Myelography Block at level Block at Block at level Block at level Block at level Block at level Block at level Block at level Block at level Block at level Block at level

Th-10 Th-6 Th-4 Th-10 Th-5

L-3 Th-9 Th-7 L-2 L-3 L-1

Discussion Rayport et al [21] have given an excellent review of the subject of hydatid cysts. According to them, hydatosis of the spine was first described by Churrier in 1807. In the literature, different percentages are given for hydatid-cyst disease causing compression of the spinal cord. The disease is seldom seen in developed countries. In Tunisia, hydatid cyst is responsible for 14% of cases of spinal-cord compression [7] and in Morocco for 4.5% [7]. In Western Europe and the United States this condition is very rare. Our 11 cases constitute 3.8% of all cases of spinal compression diagnosed, surgically treated, and hisopathologically identified during the 10 year period reported. In hydatid-cyst disease, skeletal involvement occurs in from 0 . 5 % - 3 . 1 % of cases [4,6,8, 12,15,21]. Vertebral involvement comprises 50% of this [4,7,8,12,16,19,21]. An important point is that 4 5 % o f the patients were under the age of 30. This same finding is reported in the literature [1,3,8,20]. It is known that hydatid-cyst disease can cause compression of the spinal cord [ 19,21,22,23,24]. However, except for neurological symptoms due to compression, there is no symptom or sign that is diagnostic of such disease. Generally the first complaint is radicular pain, as has been stated by Grisel and Deve [12]. An important point is that 36% of our patients had previously undergone surgery for their cysts. In the literature, 20% of patients with the condition are said to

55

have had trauma previously [7] but we were unable to verify this. In previous studies also, the incidence of paraplegia due to spinal hydatid disease was reported as being 2 5 % [22], whereas the incidence in our series was 45.4%. Additionally, the disease is said to be most commonly localized to the thoracic region [ 1,18,22]. In our series, 8 2 % of the lesions were in the thoracic area and 18% were in the lumbar area. Plain vertebral roentgenograms play an important role in the diagnosis of vertebral hydatid-cyst. Osseous changes are found in 2 7 % of cases. In plain roentgenograms, "moth-eaten" areas surrounded by sclerosis are typical. Calcifications can be found extending into the adjacent soft tissues. Tomographic examination o f the vertebral bodies has great diagnostic value for demonstrating cystic cavities and bony erosion. The Casoni and Weinberg tests, which are used for the diagnosis of hydatid disease, have no significant value if only the bones are involved [1,19,22]. In 10 of our cases (91%) the cysts were located epidurally, and in one (9%) the cysts were intradural but extramedullary. There was bony involvement in all o f our 10 cases with epidural lesions. Single epidural-cyst formation had been reported before [20], but we think that those cases manifesting a single cyst in the epidural space must be the ones in which bony involvement by multiple cysts cannot be visualized [8,21,22]. Intradural extramedullary involvement is also rarely reported in the literature [ 1,7,15,24]. Operative procedures are the treatments of choice for spinal-cord compression by hytadid cysts. The procedure chosen is usually a laminectomy. In one o f our patients, decompression with an anterior vertebrectomy and fusion was utilized and a good result was obtained. Fregerie et al [11] recommend fusion with acrylic material following anterior decompression. In our case the fusion was done with grafts obtained from the patient's rib. The patient was in good condition 3 years after the operation. In the early postoperative period, neurological improvement was seen in 7 (63%) of our patients. T h e r e was no surgical mortality. Recurrence was seen in two patients (18%). The mortality in the literature is said to range from 3% to 14.4%, and the recurrence rate from 30% to 4 0 % [1,7,25]. T h e r e is a correlation between cyst localization and recurrence. In the epidurally osseous type of cyst, one finds microvesicles diffusely spread inside the bone. Therefore, this form of the disease is referred to as "cystic disease of the vertebra" or "bone echinococcosis" [7]. In this form of the disease, these multiple cysts are easily ruptured during the surgical procedure, and recurrence is the rule [2,25]. Recurrence is very rare with the intradural extramedullary form of the disease. To prevent recurrence, it has been recommended that hypertonic saline solution be used

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Surg Neurol 1984;21:53-7

Pamir et al

T a b l e 3. Surgical Procedures, PostoperativeEarly Neurological Findings, and Subsequent Examinations in Eleven Patients with Spinal Hydatid Cysts Case no. 1

2

Operation Th9-11 laminectomy (40-50 epidural cysts) Th5-6 laminectomy (1 epidural cyst)

Postoperative early findings Paraparesis

Paraparetic; spontaneous micurition

Paraparesis

Thoracotomy for lung hydatid cyst in fifth year; reoperation of Th-8 epidural cyst in eighth year Normal in second year; reoperation of Th-6 epidural cyst in third year; normal in fifth year Paraparetic at end of first year

3

Th3-5 laminectomy (multiple epidural cysts)

Paraparesis

4

Th9-11 laminectomy (multiple epidural cysts) Th4-5 laminectomy (1 epidural cyst) L2-4 laminectomy (60-70 epidural cysts) Transthoracic vertebrectomy; anterior fusion Th6-8 laminectomy (2 epidural cysts) L1-3 laminectomy (1 subdural cyst) L2-4 laminectomy (cyst in L3 body) L 1-4 laminectomy (multiple epidural cysts)

Paraplegia

5

6

7

8

9

1()

11

during the operation. In all o f our cases the surgical area were washed with hypertonic saline, as advised. The value of this has not been established. In cases 10 and 11, m e b e n d a z o l e was used postoperatively. With this drug, g o o d results are reported in systemic Echinococcus disease [5,13,17]. The two patients have remained symptomless for the past 2 and 5 months, respectively. H o w e v e r , the overall results with this drug must be evaluated after more prolonged observation.

References 1. Acquaviva R, Tamic PM. L'echinococcose vertebromedullair6. A propos de 14 observations. Neurochirurgie 1964;10:649-50. 2. Ameli NO, Arfaa F. Hydatid disease of spine. International Congress Series, No. 293, Fifth International Congress of Neurological Surgery. Tokyo, Japan, Oct. 7-11. Amsterdam: Excerpta Medica, 1973. 3. Apt WL, Fierro JR, Calderon C, Perez C, Mujica P. Vertebral hydatid disease. J. Neurosurg 1976;44:72-6.

Follow-up examinations

Paraplegia

Paraparesis

Paraparesis

Normal at end of third year

Paraparesis

Paraparesis

Monoparesis

Normal in fifth month

Paraparesis

Normal in second month

4. Barnett LE. Hydatid disease. Aust N Z J Surg 1945;15:72. 5. Bekhti A, Schaaps JP, Carron M, Dessaint JP, Santoro F, Capron A. Treatment of hepatic hydatid disease with mebendasole: preliminary results in four cases. Br Med J 1977;2:1047-51. 6. Berkay F. Echinococcose racidienne. J Int Coil Surg 1954;22(1):35-43. 7. Bettaieb A, Khaldi T, Ben Rhouma T, Touibi S. L'echinococcose vertebro-medullarie. Neurochirurgie 1978;24:205-10. 8. Deve F. L'Echinococcose Osseuse. Monteviedo: A. Monteverde and Cia, 1948. 9- Fiennes AGTW, Thomas DGT. Combined medical and surgical treatment of spinal hydatid disease, a case report. J Neurol Neurosurg Psychiatry 1982;45:927-31. 10. Fitzpatrick SC. Hydatid disease of the lumbar vertebrae. J Bone Joint Surg 1965;47(2):286-91. 11. Fregeiro CJ, Navarro E, Pol J, Arana-Ingoez R. Vertebral hydatidosis: Treatment by vertebrectomy with substution by methylmetacrylate. International Congress Series, No. 310, Fifth International Congress of Neurological Surgery. Tokyo, Japan, Oct. 7-13 Amsterdam: Excerpta Medica, 1973. 12. Grisel P, Deve F. L'echinococcose. Rev Chit (Paris) 1929;67:375.

Spinal Hydatid Cysts

13. Kammerer WS. Medical treatment of echinococcosis. N Engl J Med 1979;301(13):727. 14. Kourias B. Apropos de 2000 cas de kystes hydatiques operSs. Br~ves considerations d'ordre statistique et chirurgical. Presse Med 1961;69(4):165-8. 15. Ley A, Marti A. Intramedullary hydatid cyst. J Neurosurg 1970;33:257-9. 16. Malloch JD. Hydatid disease of spine. Br Med J 1965;1:633. 17. Miskovitz PF, Javitt NB. Leukopenia associated with mebendasole therapy of hydatid disease. Am J Trop Med Hyg 1980;29:1356-8. 18. Morshed AA. Hydatid disease of spine. Neurochirurgia 1977;20:211-15. 19. Murray RO, Haddad F. Hydatid disease of spine. J Bone Joint Surg 1959;41:499-506.

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20. Pluchino F, Lordini S. Multiple primitive epidural spinal hydatid cysts. Acta Neurochir 1981;59:257-62. 21. Rayport M, Wisoff HS, Zaiman H. Vertebral echinocossosis. J Neurosurg 1964;21:647-59. 22. Robinson RG. Hydatid disease of spine and its neurological complications. BrJ Surg 1959;47:301-6. 23. Unger HS, Schneider LH, SherJ. Paraplegia secondary to hydatid disease. J Bone Joint Surg 1963;45A(7): 1479-84. 24. Sharma A, Kashyap V, Abraham J, Kurian S. Intradural hydatid cysts. Surg Neurol 1981;16:235-7. 25. Turtas S, Sehrbundt V, Pau A. Long term results of surgery for hydatid disease of the spine. Surg Neurol 1980; 13:468-70.