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CASE REPORT
Year : 2008  |  Volume : 3  |  Issue : 2  |  Page : 146-149
 

Craniospinal hydatidosis: Report of three cases


Department of Neurosurgery, Institute of Medical Sciences, Banaras Hindu University, Varanasi - 221 005, India

Correspondence Address:
S Shukla
Department of Neurosurgery, Institute of Medical Sciences, Banaras Hindu University, Varanasi - 221005
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/1817-1745.43643

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   Abstract 

Hydatid disease is caused by the infestation of the larvae of Taenia echinococcus. The definitive hosts of echinococcus are various carnivores, the common being the dog. All mammals (more often being sheep and cattle) are intermediate hosts. Humans are infected through the falco-oral route by the ingestion of food or milk contaminated by dog faeces that contain the ova of the parasite or by direct contact with dogs. We are reporting three cases of craniospinal hydatid cyst. First case of large intracranial hydatid cyst in a 9-year-old male child presented with holocranial headache with diminution of vision and right hemiparesis. Second case of a 34-year-old female presented with weakness, tingling, and pain right upper limb for 4 months with painless swelling right supraclavicular region. Third case of a 8-year-old child presented with low backache and paraparesis with acute retention of urine for 3 months. All the patients of craniospinal hydatidosis were managed in our department surgically.


Keywords: Cranio-spinal, hydatidosis, laminectomy


How to cite this article:
Shukla S, Trivedi A, Singh K, Sharma V. Craniospinal hydatidosis: Report of three cases. J Pediatr Neurosci 2008;3:146-9

How to cite this URL:
Shukla S, Trivedi A, Singh K, Sharma V. Craniospinal hydatidosis: Report of three cases. J Pediatr Neurosci [serial online] 2008 [cited 2019 Jul 18];3:146-9. Available from: http://www.pediatricneurosciences.com/text.asp?2008/3/2/146/43643



   Introduction Top


Hydatidosis refers to human infection with the larval form of two species of Echinococcus parasite, i.e., E. granulosus and E. multilocularis . This disease mostly found in rural and underdeveloped regions, especially agricultural and sheep-rearing communities.[1] The definitive host of Echinococcus are dogs, volves, foxes, jackals, and other carnivorous animals in which the adult parasite lives within the intestine and ova subsequently passed in stool which is ingested by intermediate host like sheep, cattle, horses, and human which then hatch into embryos (hexacanth) in the duodenum. The embryos pass through the wall of the gut into the portal system and are carried to the liver where most of the larvae are entrapped and encysted. Some may reach the lungs, and occasionally, some may pass through the capillary filter of the liver and lungs and enter into the systemic circulation. The most common sites of infestation are the liver (75%) and lungs (15%). Brain is involved in only 2-4% cases. It may affect any tissue of body except hair, teeth, and nails. [2] Vertebral hydatidosis is seen in 0.5-2% of all cases of echinococcul infestation. [3] Usually the cerebral cyst are solitary and often seen in parietal lobe [4] while spinal cyst are mostly multiple and epidural in location. [5]




   Case Reports Top


Case 1

A left-handed 9-year-old male child presented with holocranial headache for 1½ months with projectile vomiting, gradual diminution of vision, single episode right focal convulsion, and left hemiparesis. On examination patient was conscious dull with vision on right eye 6/36 left 6/6 with papilloedema grade 3 on left eye. Power 3/5 in left upper limb and lower limb at all joints. Deep tendon reflexes were brisk and plantar on left side was extensor and normal sensory examination.

Noncontrast computed tomography (CT) scan [Figure 1] revealed large hypodense nonenhancing cystic space occupying lesion without perilesional edema in right temporo-parieto-occipital region with midline shift, chest X-ray, and ultrasonography abdomen was within normal limit. Patient was operated by right fronto-temporo-parieto-occipital osteoplastic flap craniotomy with delivery of large extra-axial hydatid cyst by gradual hydrodissection in the interface between cyst and brain parenchyma.

Postoperative period was uneventful. On 3 months fellow vision was improved markedly 6/18 and power 4 + /5 in left upper and lower limb.

Case 2

A 34-year-old female presented with weakness, tingling sensation, and pain in right upper limb for 4 months. On examination grip was weak, bulk and tone was normal, power 4/5 at elbow and wrist. All deep tendon reflexes were normal except triceps which was absent. On local examination, there was painless lump in right supraclavicular region, skin was normal and mobile, temperature not raised, nonpulsatile, noncompressible with absent cough reflex.

T1W magnetic resonance imaging (MRI) revealed isointense mass lesion in supraclavicular region up to apex of right lung extending into neural foramina of right C 6-7 with mild encroachment in spinal canal. On T2W MRI [Figure 2] mass was hyperintense. Patient was approached through right supraclavicular region with identification of lower cord of brachial plexus which was tented by multiple small transparent, cystic lesion [Figure 3]. The hundreds of daughter cysts excised completely and the cavity was irrigated with saline [Figure 4]. Postoperative period was uneventful and patient became asymptomatic at the time of discharge.

Case 3

A 8-year-old male child presented with low backache, weakness of both lower limbs, and acute retention of urine for 3 months. On examination, he had paraparesis grade 3/5 in both lower limbs at knee and ankle. Knee, ankle reflex absent, and plantar was nonelicitible on both side. T1W MRI revealed isointense nonenhancing cystic space occupying lesion at cauda-equina below conus [Figure 5]. On T2W MRI lesion was hyperintense [Figure 6].

Patient was operated with laminectomy from D12-L2, intradural transparent cystic lesion adherent to nerve roots were found and separated [Figure 7] and [Figure 8]. Postoperative period was uneventful. On 3 months follow-up, patient can walk with support with normal bladder function.

All these patients were put on albendazole 10 mg/kg/d for 3 months.


   Discussion Top


Intracranial hydatid disease is rare, with a reported incidence of 1-2% in all the cases with hydatid disease. [6] Cerebral hydatid disease is more common in the pediatric population. [7],[8] The history of direct contact with dogs is not available in all the cases as the infection can be acquired by eating contaminated food and milk.

Intracranial hydatid cysts are more frequently located in the supratentorial compartment. The parietal lobe is the most common site. All the four cases reported by Dharker et al. [9] and three out of five cases of intracerebral hydatid cysts as reported by Balasubramanium et al, [10] had parietal lobe involvement. The other less common sites are skull, [4] cavernous sinus, [11] eye ball, [12] pons, [13] cerebellum, and ventricles. [8] The growth rate of the hydatid cysts of the brain has been variably reported between 1.5 and 10 cm/year.

Solitary intracranial hydatid cysts are common; multiple intracranial cysts are rare. Onal et al , [14] found only three cases of multiple cysts in their series of 33 cases and Lunardi et al , [15] found two cases in their series of 12 cases. The patients with intracranial hydatid cysts usually present with focal neurological deficit and features of raised intracranial pressure; the latter may be due to the large size or due to interference with pathway of the cerebrospinal fluid (CSF) flow. Erashin et al , [6] observed that 18 out of 19 cases presented with raised intracranial pressure. MR and CT scans characteristically show hydatid cyst as a spherical, well-defined, nonenhancing cystic lesion without peripheral edema. [16],[17] On CT and MR scan, the fluid density is found to be generally equal to that of CSF. A fine rim of peripheral enhancement with perilesional edema may be seen in the presence of active inflammation. [17] In our case, the patient presented with headache with focal neurological deficit, and papilledema with the diminution of vision. Long-term follow-up confirms that intracranial hydatid cysts should always be surgically removed without rupture; the outcome remains excellent in these cases. Medical treatment with albendazole seems to be beneficial both pre- and postoperatively. Novel diagnostic methodologies such as MR spectroscopy and MR diffusion-weighted imaging might help in the diagnosis of intracranial hydatid cysts. [18]

The primary cysts are formed as a result of direct infestation of the larvae in the brain without the demonstrable involvement of other organs. In primary multiple cysts, each cyst has a separate pericyst with brood capsule scolices, and they originate from multiple larvae that affect brain after crossing the gastrointestinal tract, liver, lungs, and right side of heart without affecting them. The primary cysts are fertile as they contain scolices and brood capsules; hence, the rupture of primary cyst can result in recurrence. The secondary multiple cysts results from spontaneous, traumatic, or surgical rupture of the primary intracranial hydatid cyst, and they lack brood capsule and scolices. The secondary intracranial hydatid cysts are, therefore, infertile and the resultant risk of recurrence after their rupture is negligible.

Various surgical techniques commonly for used for removal of the cysts are summarized by Arana Iniquez [19] and include direct puncture and aspiration of the cyst fluid through a small hole in the cyst wall, and expulsion of the cyst through a small cortical incision over cyst using insuffalation of air in the contralateral ventricle. The most popular technique, however, is to use irrigation saline with mild force between the cyst wall and brain interface to deliver the cyst intact. This is often possible because the adhesions around the cyst wall are minimal. We performed this procedure in our case, as shown in operative slides.

Bone involvement by hydatid is uncommon only 0.5-1% of all hydatid disease cases involve spine. The most common sign and symptoms include paraparesis 62%, paraplagia 26%, backache and radicular pain 55%, sensory loss 36%, and sphincter disturbance in 3%.

Primary hydatid cysts of the spinal cord are extremely rare and in almost every case spinal cord compression is caused secondarily by hydatid disease of the vertebrae. Extradural hydatid cysts without vertebral involvement have been reported. [20] Medjek et al, [21] reported an intradural spinal hydatid cyst in the thoracic region and Akhan et al, [22] one in a child. Natarajan [3] reported a spinal hydatid cyst causing compression of the cauda equina.

CT scan with myelography or MR is investigation of choice for spinal hydatid cyst. Postoperative MRI is invaluable for determining the completeness of resection as well as for documenting any recurrent or newly developing cysts. Surgery is the most common initial treatment with total removal of the cyst, the primary goal. Laminectomy with simple decompression is used most frequently, yet it is not without accompanying risk.

Only a few reports are available that mention the efficacy of albendazole therapy. Isolated case reports [2],[17] showed complete disappearance of the multiple intracranial hydatid cysts with albendazole therapy with a daily dose of 10 mg/kg, taken three times a day for 4 months. Erashin et al, [6] reported better effectiveness of the drug therapy in recurrent cases and in cases with rupture at the surgery. We have advised the administration of albendazole 10 mg/kg postoperatively for 1 month.

 
   References Top

1.Markell EK, Voge M. Medical parasitology, 5 th ed. Philadelphia: Saunders; 1981.  Back to cited text no. 1    
2.Singounas EG, Leventis AS, Sakas DE, Hadley DM, Lampadarios DA, Karvounis PC. Successful treatment of intracerebral hydatid cysts with albendazole: Case report and review of the literature. Neurosurgery 1992;31:571-4.  Back to cited text no. 2  [PUBMED]  [FULLTEXT]
3.Natarajan MA. Case of spinal hydatid cyst causing cauda equina compression. Indian J Surg 1974;36:44.  Back to cited text no. 3    
4.Boles DM. Cerebral echinococciasis. Surg Neurol 1981;16:280-2.  Back to cited text no. 4  [PUBMED]  
5.Pamir MN, Akalan N, Ozgen T, Erbengi A. Spinal hydatid cysts. Surg Neurol 1984;21:53-7.  Back to cited text no. 5  [PUBMED]  
6.Erashin Y, Mutluer S, Guzelbag H. Intracranial hydatid cysts in children. Neurosurgery 1993;33:219-25.  Back to cited text no. 6    
7.Dharker SR. Hydatid disease. In: Text book of neurosurgery, 2 nd ed. In: Ramamurthi B, Tandon PN, editors. Churchill Livingstone: New Delhi; 1996. p. 535,44.  Back to cited text no. 7    
8.Jinsenez-Mejias ME, Castillo-Ojeda E, Cuello-Contreras JA, Garcia Gutirrez D, Romero Arce J, Pachon Diaz J. Cerebral hydatidosis: Analysis of a series of 23 cases. Med Clin (Barc) 1991;97:125-32.  Back to cited text no. 8    
9.Dharker SR, Dharker RS, Vaishya ND, Sharma ML, Chaurasia BD. Cerebral hydatid cysts in central India. Surg Neurol 1977;8:31-4.  Back to cited text no. 9  [PUBMED]  
10.Balasubramanium V, Ramanujani PB, Ramamurthi B. Hydatid disease of the nervous system. Neurol India 1970;18:92-5.  Back to cited text no. 10    
11.Rivierez M, el-Azhari A, el-Tantaoui M, Alaoui M. Hydatid cyst of cavernous sinus (a case). Neurochirurgie 1992;38:46-9.  Back to cited text no. 11    
12.Sinav S, Demirei A, Sinav B, Oge F, Sullu Y, Kandemir B. A primary intraocular hydatid cyst. Acta Ophthalmol (Copenh) 1991;69:802-4.  Back to cited text no. 12    
13.Abbassioun K, Amirjamshidi A, Moinipoor MT. Hydatid cyst of the pons. Surg Neurol 1986;26:207-300.  Back to cited text no. 13    
14.Onal C, Barlas O, Orakdogen M, Hepgul K, Izgi N, Unal F. Three unusual cases of intracranial hydatid cyst in paediatric age group. Pediatr Neurosurg 1997;26:208-13.  Back to cited text no. 14    
15.Lunardi P, Missori P, Di-Lorenzo N, Fortuna A. Cerebral hydandosis in childhood: A retrospective survey with emphasis, long term follow up. Neurosurgery 1991;29:515-8.  Back to cited text no. 15    
16.Golemati B, Lakiotis G, Persidou-Golemati P, Bonatsos G. Albendazole in the conservative treatment of multiple hydatid disease. Mt Sinai J Med 1989;56:53-5.  Back to cited text no. 16    
17.Todorov T Vutova K, Petkov D, Balkanski C. Alberidzole treatment of multiple cerebral hydatid cysts: A case report. Trans R Soc Trop Med Hyg 1988;82:150-2.  Back to cited text no. 17    
18.Ciurea AV, Fountas KN, Consan TC, Machinis TO, Kapsalaki EZ, Fezouhdis NI, et al. Long-term surgical outcome in patients with intracranial hydatid cyst. Acta Neurochir (Wien) 2006;148:421-6.  Back to cited text no. 18    
19.Arana Iniquez R. Echinococcus. Infection of the nervous system. In: Hand Book of Clinical Neurology, Part 3, Vinken P3, Bruyn OW, editors. Elsevier/North Holland Bi, Medical Press: Amsterdam; 1978, 175-208.  Back to cited text no. 19    
20.Sinha VD, Mathur PP, Dharker SR. Spinal extradural hydatid cyst. Neurol India 1990;38:222.  Back to cited text no. 20    
21.Medjek L, Zenini S, Hammoum S, Hartani M. Intradural hydatidosis of the thoracic spine. Ann Radiol (Paris) 1991;34:251-5.  Back to cited text no. 21  [PUBMED]  
22.Akhan O, Dinηer A, Saatηi I, Gülekon N, Besim A. Spinal intradural hydatid cyst in a child. Br J Radiol 1991;64:465-6.  Back to cited text no. 22    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7], [Figure 8]


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