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Dumbbell shaped spinal hydatidosis Krishna Chaitanya Joshi, Aniruddh T. Jagannath, Ravi Gopal Varma Department of Neurosurgery, M. S. Ramaiah Institute of Neurosciences, Bengaluru, Karnataka, India Sir, A 28-year-old male patient from Iraq presented with progressively increasing pain on the left side of chest since past 2 months and weakness of lower limbs since 2 weeks. Neurological examination revealed spastic paraparesis with motor power of 4/5. Local examination of spine revealed tenderness over the spinous process of T5 and T6 vertebrae. Chest X-ray revealed homogenous mediastinal opacity on the left side at the level of T5-6 and erosion of the head of left fifth rib. Contrast enhanced magnetic resonance imaging revealed 4 × 5 cm cystic lesion in the posterior mediastinum left paravertebral region with extension into the spinal canal through T5-6 foramen. The intra spinal extradural part had multiple septations and had a contrast enhancing cyst wall [Figure 1]. He underwent T5-6 laminectomy and at operation there were multiple cysts in the extradural space with a large cyst extending into the posterior mediastinum on the left side. The cavity was irrigated with 3 % hypertonic saline. During resection of the pericyst we encountered a small breech in the parietal pleura, which was sutured. The histopathological examination of cysts revealed them to be Echinoccous granulosus. Hydatid disease is caused by the larval stage of the cestode, E. Granulosus and is highly prevalent in countries with temperate climates.[1] The lifecycle of E. granulosus involves two hosts. The definitive host is a dog or other related carnivore where the adult parasite lodges in the proximal small bowel and the eggs are shed in the feces. The intermediate host, usually a sheep (or some other herbivore), gets infected when grazing on contaminated ground. The embryo (oncosphere) hatches, penetrates the intestinal mucosa, develops into the characteristic vesicular metacestode on reaching a suitable anatomical site. When the definitive host feeds on infected viscera, the cycle is complete.[2] Humans are accidental hosts. Access this article online Quick Response Code:
Website: www.neurologyindia.com PMID: *** DOI: 10.4103/0028-3886.128363
Neurology India | Jan-Feb 2014 | Vol 62 | Issue 1
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Figure 1: (a) Chest X-ray showing a homogenous density with smooth borders in the mediastinum on the left side (b) T2 weighted axial image at level of T5 showing a hyperintense mass on left paravertebral region extending into the spinal canal, displacing the thecal sac (c) T2 HalfFourier acquisition with single-shot turbo spin-echo sequence showing a multiple cystic lesions extending through the foramen (d) T1 contrast coronal image showing wall enhancement
Spinal hydatidosis, involvement of the spinal cord, the spine, or both structures, is associated with a high degree of morbidity and mortality and the prognosis has often been compared with that of malignancies.[3-5] Extra-visceral hydatid disease is generally thought to evolve from arterial dissemination of the oncospheres and the other proposed route is venous, through porto-vertebral shunts and the retrograde passage of the parasite from the inferior vena cava to retroperitoneal and epidural venous plexuses.[6] Mediastinal hydatid cyst with intraspinal extension is an extremely rare entity, less than ten cases have been reported until date.[7] Extension of the lesion through the spinal canal gives them the unique dumbbell shape.[8] The differential diagnosis of dumbbell lesion include benign/malignant peripheral nerve sheath tumors, plasmacytoma, chondrosarcoma, chondroid chordoma, superior sulcus tumor, metastasis and non-neoplastic lesions such as infectious process (tuberculosis, hydatid cyst), aneurysmal bone cyst, synovial cyst, traumatic pseudomeningocele, arachnoid cyst and hemangiomas. Mediastinal hydatid cysts with intra spinal extensions can lead to destruction of the vertebral body and the adjacent rib. Braithwaite and Lees[9] have anatomically classified spinal hydatidosis into five categories: Type 1 – intramedullary, type 2 – 113
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intradural extramedullary, type 3 – extradural intraspinal, type 4 – vertebral and type 5 – paravertebral or dumbbell shaped. Mediastinal hydatid should always be considered in the differential diagnosis of cystic lesions with intra spinal extension, especially in endemic areas and serological testing should be considered pre-operatively. The best treatment for spinal hydatidosis is surgical excision of the germinative membrane and pericyst. Pre-operative treatment with benzimidazoles has been reported to soften the cysts and to reduce intracystic pressure, enabling the surgeon to remove the endocyst more easily. It is recommended for cases in which spillage of protoscoleces may have occurred during surgery to initiate post-operative chemotherapy with albendazole (ABZ) or mebendazole (MBZ) immediately after the operation for at least 1 month (ABZ) or 3 months (MBZ).[10] Address for correspondence: Dr. Krishna Chaitanya Joshi, Department of Neurosurgery, M. S. Ramaiah Institute of Neurosciences, MSRIT Post, New BEL Road, Bengaluru, Karnataka, India. E-mail: [email protected]
Worldwide epidemiology of liver hydatidosis including the Mediterranean area. World J Gastroenterol 2012;18:1425-37. 2. Schantz PM, PB, E. In: Guerrant RL, DH, Weller PF, editors. Echinococcosis. 2nd ed. Philadelphia: Churchill Livingstone (Elsevier); 2006. 3. Kammerer WS. Echinococcosis affecting the central nervous system. Semin Neurol 1993;13:144-7. 4. Ozdemir HM, Ogün TC, Tasbas B. A lasting solution is hard to achieve in primary hydatid disease of the spine: Long-term results and an overview. Spine (Phila Pa 1976) 2004;29:932-7. 5. Pedrosa I, Saíz A, Arrazola J, Ferreirós J, Pedrosa CS. Hydatid disease: Radiologic and pathologic features and complications. Radiographics 2000;20:795-817. 6. Pamir MN, Ozduman K, Elmaci I. Spinal hydatid disease. Spinal Cord 2002;40:153-60. 7. Ranganadham P, Dinakar I, Sundaram C, Ratnakar KS, Vivekananda T. Posterior mediastinal paravertebral hydatid cyst presenting as spinal compression. A case report. Clin Neurol Neurosurg 1990;92:149-51. 8. Kilic D, Erdogan B, Sener L, Sahin E, Caner H, Hatipoglu A. Unusual dumbbell tumours of the mediastinum and thoracic spine. J Clin Neurosci 2006;13:958-62. 9. Braithwaite PA, Lees RF. Vertebral hydatid disease: Radiological assessment. Radiology 1981;140:763-6. 10. Pawłowski ZS, Eckert J, Vuitton DA, Ammann RW, Kern P, Craig PS, et al. Echinococcosis in humans: Clinical aspects, diagnosis and treatment. In: Eckert J, Johannes Eckert, Meslin F-X, Pawłowsk ZS, editors. Paris: World Organisation for Animal Health (Office International des Epizooties) and World Health Organization; 2001.
References
How to cite this article: Joshi KC, Jagannath AT, Varma RG. Dumbbell shaped spinal hydatidosis. Neurol India 2014;62:113-4.
1.
Source of Support: Nil, Conflict of Interest: None declared.
114
Grosso G, Gruttadauria S, Biondi A, Marventano S, Mistretta A.
Neurology India | Jan-Feb 2014 | Vol 62 | Issue 1
Copyright of Neurology India is the property of Medknow Publications & Media Pvt. Ltd. and its content may not be copied or emailed to multiple sites or posted to a listserv without the copyright holder's express written permission. However, users may print, download, or email articles for individual use.
Neuroimage
Dumbbell shaped spinal hydatidosis Krishna Chaitanya Joshi, Aniruddh T. Jagannath, Ravi Gopal Varma Department of Neurosurgery, M. S. Ramaiah Institute of Neurosciences, Bengaluru, Karnataka, India Sir, A 28-year-old male patient from Iraq presented with progressively increasing pain on the left side of chest since past 2 months and weakness of lower limbs since 2 weeks. Neurological examination revealed spastic paraparesis with motor power of 4/5. Local examination of spine revealed tenderness over the spinous process of T5 and T6 vertebrae. Chest X-ray revealed homogenous mediastinal opacity on the left side at the level of T5-6 and erosion of the head of left fifth rib. Contrast enhanced magnetic resonance imaging revealed 4 × 5 cm cystic lesion in the posterior mediastinum left paravertebral region with extension into the spinal canal through T5-6 foramen. The intra spinal extradural part had multiple septations and had a contrast enhancing cyst wall [Figure 1]. He underwent T5-6 laminectomy and at operation there were multiple cysts in the extradural space with a large cyst extending into the posterior mediastinum on the left side. The cavity was irrigated with 3 % hypertonic saline. During resection of the pericyst we encountered a small breech in the parietal pleura, which was sutured. The histopathological examination of cysts revealed them to be Echinoccous granulosus. Hydatid disease is caused by the larval stage of the cestode, E. Granulosus and is highly prevalent in countries with temperate climates.[1] The lifecycle of E. granulosus involves two hosts. The definitive host is a dog or other related carnivore where the adult parasite lodges in the proximal small bowel and the eggs are shed in the feces. The intermediate host, usually a sheep (or some other herbivore), gets infected when grazing on contaminated ground. The embryo (oncosphere) hatches, penetrates the intestinal mucosa, develops into the characteristic vesicular metacestode on reaching a suitable anatomical site. When the definitive host feeds on infected viscera, the cycle is complete.[2] Humans are accidental hosts. Access this article online Quick Response Code:
Website: www.neurologyindia.com PMID: *** DOI: 10.4103/0028-3886.128363
Neurology India | Jan-Feb 2014 | Vol 62 | Issue 1
a
b
c
d
Figure 1: (a) Chest X-ray showing a homogenous density with smooth borders in the mediastinum on the left side (b) T2 weighted axial image at level of T5 showing a hyperintense mass on left paravertebral region extending into the spinal canal, displacing the thecal sac (c) T2 HalfFourier acquisition with single-shot turbo spin-echo sequence showing a multiple cystic lesions extending through the foramen (d) T1 contrast coronal image showing wall enhancement
Spinal hydatidosis, involvement of the spinal cord, the spine, or both structures, is associated with a high degree of morbidity and mortality and the prognosis has often been compared with that of malignancies.[3-5] Extra-visceral hydatid disease is generally thought to evolve from arterial dissemination of the oncospheres and the other proposed route is venous, through porto-vertebral shunts and the retrograde passage of the parasite from the inferior vena cava to retroperitoneal and epidural venous plexuses.[6] Mediastinal hydatid cyst with intraspinal extension is an extremely rare entity, less than ten cases have been reported until date.[7] Extension of the lesion through the spinal canal gives them the unique dumbbell shape.[8] The differential diagnosis of dumbbell lesion include benign/malignant peripheral nerve sheath tumors, plasmacytoma, chondrosarcoma, chondroid chordoma, superior sulcus tumor, metastasis and non-neoplastic lesions such as infectious process (tuberculosis, hydatid cyst), aneurysmal bone cyst, synovial cyst, traumatic pseudomeningocele, arachnoid cyst and hemangiomas. Mediastinal hydatid cysts with intra spinal extensions can lead to destruction of the vertebral body and the adjacent rib. Braithwaite and Lees[9] have anatomically classified spinal hydatidosis into five categories: Type 1 – intramedullary, type 2 – 113
[Downloaded free from http://www.neurologyindia.com on Thursday, March 13, 2014, IP: 202.177.173.189] || Click here to download free Android application for this journal Joshi, et al.: Spinal hydatidosis
intradural extramedullary, type 3 – extradural intraspinal, type 4 – vertebral and type 5 – paravertebral or dumbbell shaped. Mediastinal hydatid should always be considered in the differential diagnosis of cystic lesions with intra spinal extension, especially in endemic areas and serological testing should be considered pre-operatively. The best treatment for spinal hydatidosis is surgical excision of the germinative membrane and pericyst. Pre-operative treatment with benzimidazoles has been reported to soften the cysts and to reduce intracystic pressure, enabling the surgeon to remove the endocyst more easily. It is recommended for cases in which spillage of protoscoleces may have occurred during surgery to initiate post-operative chemotherapy with albendazole (ABZ) or mebendazole (MBZ) immediately after the operation for at least 1 month (ABZ) or 3 months (MBZ).[10] Address for correspondence: Dr. Krishna Chaitanya Joshi, Department of Neurosurgery, M. S. Ramaiah Institute of Neurosciences, MSRIT Post, New BEL Road, Bengaluru, Karnataka, India. E-mail: [email protected]
Worldwide epidemiology of liver hydatidosis including the Mediterranean area. World J Gastroenterol 2012;18:1425-37. 2. Schantz PM, PB, E. In: Guerrant RL, DH, Weller PF, editors. Echinococcosis. 2nd ed. Philadelphia: Churchill Livingstone (Elsevier); 2006. 3. Kammerer WS. Echinococcosis affecting the central nervous system. Semin Neurol 1993;13:144-7. 4. Ozdemir HM, Ogün TC, Tasbas B. A lasting solution is hard to achieve in primary hydatid disease of the spine: Long-term results and an overview. Spine (Phila Pa 1976) 2004;29:932-7. 5. Pedrosa I, Saíz A, Arrazola J, Ferreirós J, Pedrosa CS. Hydatid disease: Radiologic and pathologic features and complications. Radiographics 2000;20:795-817. 6. Pamir MN, Ozduman K, Elmaci I. Spinal hydatid disease. Spinal Cord 2002;40:153-60. 7. Ranganadham P, Dinakar I, Sundaram C, Ratnakar KS, Vivekananda T. Posterior mediastinal paravertebral hydatid cyst presenting as spinal compression. A case report. Clin Neurol Neurosurg 1990;92:149-51. 8. Kilic D, Erdogan B, Sener L, Sahin E, Caner H, Hatipoglu A. Unusual dumbbell tumours of the mediastinum and thoracic spine. J Clin Neurosci 2006;13:958-62. 9. Braithwaite PA, Lees RF. Vertebral hydatid disease: Radiological assessment. Radiology 1981;140:763-6. 10. Pawłowski ZS, Eckert J, Vuitton DA, Ammann RW, Kern P, Craig PS, et al. Echinococcosis in humans: Clinical aspects, diagnosis and treatment. In: Eckert J, Johannes Eckert, Meslin F-X, Pawłowsk ZS, editors. Paris: World Organisation for Animal Health (Office International des Epizooties) and World Health Organization; 2001.
References
How to cite this article: Joshi KC, Jagannath AT, Varma RG. Dumbbell shaped spinal hydatidosis. Neurol India 2014;62:113-4.
1.
Source of Support: Nil, Conflict of Interest: None declared.
114
Grosso G, Gruttadauria S, Biondi A, Marventano S, Mistretta A.
Neurology India | Jan-Feb 2014 | Vol 62 | Issue 1
Copyright of Neurology India is the property of Medknow Publications & Media Pvt. Ltd. and its content may not be copied or emailed to multiple sites or posted to a listserv without the copyright holder's express written permission. However, users may print, download, or email articles for individual use.
