A novel surgical technique for hydatid cyst involving cervicothoracic anterior epidural space

Bharat R Dave; Degulmadi Devanand; Ganesh Deshmukh

doi:10.4103/isj.isj_17_18

CASE REPORT

Year : 2019 | Volume : 2 | Issue : 1 | Page : 99-101

A novel surgical technique for hydatid cyst involving cervicothoracic anterior epidural space

Bharat R Dave, Degulmadi Devanand, Ganesh Deshmukh
Department of Spine Surgery, Stavya Spine Hospital and Research Institute Pvt. Ltd, Ahmedabad, Gujarat, India

Date of Web Publication

11-Jan-2019

Correspondence Address:
Dr. Degulmadi Devanand
Stavya Spine Hospital and Research Institute Pvt. Ltd., Mithakali, Gujarat, Ahmedabad
India

Source of Support: None, Conflict of Interest: None

DOI: 10.4103/isj.isj_17_18

Abstract

Spinal hydatid cyst comprises <1% of the total cases of hydatid disease. There is very little literature on the involvement of anterior epidural space by hydatid cyst and its management. This report presents a unique presentation of spinal hydatidosis in cervicothoracic anterior epidural space and a novel technique in surgical management.

Keywords: Anterior epidural space, cervicothoracic spine, hydatid cyst

How to cite this article:
Dave BR, Devanand D, Deshmukh G. A novel surgical technique for hydatid cyst involving cervicothoracic anterior epidural space. Indian Spine J 2019;2:99-101

How to cite this URL:
Dave BR, Devanand D, Deshmukh G. A novel surgical technique for hydatid cyst involving cervicothoracic anterior epidural space. Indian Spine J [serial online] 2019 [cited 2021 Nov 29];2:99-101. Available from: https://www.isjonline.com/text.asp?2019/2/1/99/249893

Introduction

Hydatid disease or hydatidosis is a widespread zoonosis caused by Echinococcus granulosus.^[1] Bone involvement is rare and is reported in 0.5%–4% of the cases.^[2] Of the areas involved in the spine, the thoracic spine is most commonly involved followed by the lumbar spine.^[3],[4] Involvement of multiple sites in the spine is uncommon and seldom reported. Diagnosis in such presentations is aided with neuroimaging and immunohistochemical modalities. Surgical approach to such rare cases is a challenge to the treating surgeon. We report a case treated with novel surgical technique that presented to us with extensive involvement of anterior epidural space in the cervicothoracic area with neurological deficit.

Case Report

A 35-year-old female was admitted to our hospital with neck pain, progressive weakness of both the lower limbs and bilateral handgrip weakness with paresthesias Grade 3 power, non-walker. No other medical history suggestive of lung or liver disease was present. A detailed general physical examination revealed no abnormality. On clinical examination, power was Grade 3/5 in both the lower limbs and loss of handgrip power in the upper limbs. Deep tendon reflexes were exaggerated, and sensations were decreased below C6. Routine blood investigations were within normal limits. Radiographs of the cervical and thoracic spine were normal. Magnetic resonance imaging (MRI) spine showed multiple well-defined extradural multilocular cystic lesions, hypointense on T1 and hyperintense on T2, occupying the anterior epidural space from C5 to T11 suggestive of hydatid cyst [Figure 1]a. Ultrasound sonography abdomen showed two well-defined cystic lesions in the liver with partial calcification in the wall suggestive of hydatid disease.

Figure 1: (a) Preoperative magnetic resonance imaging T2 images showing multiple well-defined extradural cystic lesions occupying the anterior epidural space from C5 to D11. (b) Intraoperative findings of multiple cysts filling up the corpectomy defect with catheter in situ showing 25 mm mark. (c) Cysts after aspiration (d) Postoperative magnetic resonance imaging showing expanded anterior epidural space devoid of cysts

Click here to view

Surgical planning, in this case, was most challenging. Available options were posterior laminectomy and approaching anterior epidural space, anterior thoracotomy with corpectomy of dorsal vertebra and evacuation of epidural space, and lastly, cervical corpectomy for clearance of the epidural space. After detailed counseling and consent of the patient, we opted for the cervical corpectomy for approaching the anterior epidural space and aspiration of cystic material. A total of 1 g Solumedrol intravenously was given as a prophylactic measure to avoid anaphylactic reactions that are known to occur with cyst evacuation. Following C6 corpectomy, a collection of cysts was noted in the anterior epidural space. A 6G infant feeding tube with 2 mm outer diameter (with radiopaque line) was passed into the anterior epidural space under image guidance, and wash was given with 0.9% saline [Figure 1]b. Tube was passed with caution so as not to damage or cause any cystic material spill over. Multiple cysts were seen to be filling up the corpectomy defect [Figure 1]b and [Figure 1]c. Tube was further passed all along up to T11, confirmed by the radiopaque dye (Omnipaque), and thorough irrigation suction was done until no more cysts were seen coming out in the corpectomy defect. Anterior cervical fusion was performed with a mesh cage filled with bone graft and plating from C5 to 7 for stabilization. Patient's neurology showed signs of improvement within 24 h of surgery.

Histopathological examination was confirmative of hydatid cyst. Tablet albendazole 400 mg daily was started and continued for 6 months. Remarkable clinical improvement was seen in the postoperative period, and follow-up till 1 year showed no radiological recurrence [Figure 1]d. There was a significant change in the dimensions of cord diameter [Table 1].

Table 1: Change in width of the spinal cord on sagittal T2-magnetic resonance imaging image (mm)

Click here to view

Discussion

Spinal hydatid disease manifests itself through symptoms and signs related to compression of the cysts on neighboring structures; no specific pathognomonic symptoms or signs exist.^[1],[5] Due to its uncommon presentation, unless the clinician includes spinal hydatid disease as part of the differential diagnosis this potentially curable disease is usually missed.^{[6],[7],[8],[9]} In such uncommon presentations, other conditions such as Koch's spine, arachnoid cyst, pyogenic infection, mycosis, and spinal abscess require proper evaluation as they can closely mimic spinal hydatid cyst. Although many spinal hydatid cyst cases are reported in the literature, the involvement of anterior epidural space is uncommon and rarely reported. The present case reports a unique presentation of spinal hydatid cyst with extensive involvement of the anterior epidural space from cervical (C4) to dorsal (T11) spine.

In some patients, the spinal hydatid cysts can grow to enormous size and clinically remain asymptomatic for years.^[10],[11] Our patient presented late with neurological deficit and extensive involvement of the anterior epidural space of cervical as well as the thoracic spine. Majority of the cases (>90%) are managed by laminectomy; however, such an extensive involvement in the inaccessible areas such as anterior epidural space needs unique planning. In the present case, a novel technique in the surgical management of such uncommon presentation is discussed. Anterior cervical corpectomy was planned, as the patient presented to us with increasing handgrip weakness and radiological involvement of collection extending into the cervical spine.

As rupture of cysts during surgery of spinal hydatid cyst is a central problem, most surgeons advocate the use of scolicidal agents to irrigate the operative field although the efficacy of this measure remains unproven, and potentially deleterious effects on neural tissue have to be considered.^{[11],[12],[13]} We have used 0.9% saline for irrigation of the cavity, and scolicidal agents were not used for fear of unrecognized dural injury as passing the tube distally for wash was a blind procedure. As the anterior epidural space is very narrow and chances of cyst rupture are very high as published in the literature, we have taken all the possible measures to avoid rupture of cysts.^[4],[14] Gentle wash was given, and the cysts were aspirated with wide bore suction cannula to avoid rupture of cysts. Furthermore, we have used a narrow 6G infant feeding tube with 2 mm outer diameter when the available anterior epidural space was 2.6 mm as measured on MRI. It can be argued that the potential complication of this technique is neurological deterioration, but this was the best available option in such a rare presentation.

Besides leading to disease recurrence, the spillage of cyst content during surgery may provoke various hypersensitivity reactions ranging from pruritus, rash, edema, bronchospasm, and gastrointestinal symptoms to hypotension and fatal anaphylactic shock.^[15] We have used intravenous glucocorticosteroids (1 g Solumedrol) to prevent anaphylactic shock. In the series published by Pamir et al., the neurological improvement was seen in 63% of the cases and recurrence in 18%.^[1] Overall, a recurrence rate of 30%–40% is described.^[16] There was a significant neurological recovery in our case, and the patient started walking with support within a week. In the follow-up of 1 year, there was no radiological recurrence as noted on the follow-up MRI.

Albendazole is the preferred antihelminthic agent in the postoperative treatment of hydatid disease, but the duration of treatment is controversial.^[17] Albendazole acts by blocking glucose uptake and depleting the glycogen stores of the parasite. Medical management in our case included albendazole after histopathological confirmation for 6 months with monthly checkup of complete blood picture and liver function tests.

Conclusion

We report a rare case of spinal hydatid disease with extensive anterior epidural space involvement in which we employed a novel surgical technique. Although our technique has a risk of neurological worsening, in our view it was the only option available in the management of such rare presentation.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

References

1.	Pamir MN, Akalan N, Ozgen T, Erbengi A. Spinal hydatid cysts. Surg Neurol 1984;21:53-7.
2.	Charles RW, Govender S, Naidoo KS. Echinococcal infection of the spine with neural involvement. Spine (Phila Pa 1976) 1988;13:47-9.
3.	Song X, Liu D, Wen H. Diagnostic pitfalls of spinal echinococcosis. J Spinal Disord Tech 2007;20:180-5.
4.	Işlekel S, Erşahin Y, Zileli M, Oktar N, Oner K, Ovül I, et al. Spinal hydatid disease. Spinal Cord 1998;36:166-70.
5.	Berk C, Ciftçi E, Erdoğan A. MRI in primary intraspinal extradural hydatid disease: Case report. Neuroradiology 1998;40:390-2.
6.	Ndondo AP, Fieggen G, Wilmshurst JM. Hydatid disease of the spine in South African children. J Child Neurol 2003;18:343-6.
7.	Yilmaz N, Ozgocmen S, Kocakoc E, Kiris A. Primary hydatid disease of sacrum affecting the sacroiliac joint: A case report. Spine (Phila Pa 1976) 2004;29:E88-90.
8.	Ergin A, Toker T, Yanarates O, Kurt E, Guzeldemir ME. A typical low-back pain caused by an atypical etiology. Reg Anesth Pain Med 2007;32:89-92.
9.	Layadi F, Boubrik M, Aït El Qadi A, Aït Benali S. Primary sacral epidural hydatid cyst: A case report. J Radiol 2005;86:1040-2.
10.	Joshi N, Hernandez-Martinez A, Seijas-Vazquez R. Primary sacral hydatid cyst. A case report. Acta Orthop Belg 2007;73:674-7.
11.	Govender TS, Aslam M, Parbhoo A, Corr P. Hydatid disease of the spine. A long-term followup after surgical treatment. Clin Orthop Relat Res 2000;(378):143-7.
12.	Pamir MN, Ozduman K, Elmaci I. Spinal hydatid disease. Spinal Cord 2002;40:153-60.
13.	Turgut M. Hydatid disease of the spine: A survey study from Turkey. Infection 1997;25:221-6.
14.	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.
15.	Neumayr A, Troia G, de Bernardis C, Tamarozzi F, Goblirsch S, Piccoli L, et al. Justified concern or exaggerated fear: The risk of anaphylaxis in percutaneous treatment of cystic echinococcosis-a systematic literature review. PLoS Negl Trop Dis 2011;5:e1154.
16.	Turtas S, Viale ES, Pau A. Long-term results of surgery for hydatid disease of the spine. Surg Neurol 1980;13:468-70.
17.	Fiennes AG, Thomas DG. Combined medical and surgical treatment of spinal hydatid disease: A case report. J Neurol Neurosurg Psychiatry 1982;45:927-30.