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Year : 1999 | Volume
: 47
| Issue : 1 | Page : 79-81 |
Cervical intradural extramedullary bronchiogenic cyst.
Rao GP, Bhaskar G, Reddy PK
Department of Neurosurgery, Osmania Medical College/Osmania General Hospital, Hyderabad, Andhra Pradesh, India.
Correspondence Address: Department of Neurosurgery, Osmania Medical College/Osmania General Hospital, Hyderabad, Andhra Pradesh, India.
How to cite this article: Rao G P, Bhaskar G, Reddy P K. Cervical intradural extramedullary bronchiogenic cyst. Neurol India 1999;47:79-81 |
Spinal bronchiogenic cysts are congential in origin. These are grouped under enterogenic cysts. The present case is of an enterogenous cyst(bronchiogenic type) located at high cervical region, a rare site. Spinal bronchiogenic cysts are congenital in origin. These cysts are grouped under enterogenous cysts. Enterogenous cysts have lining epithelium similar to the structures that originate from primitive foregut, i.e. the oesophagus, the respiratory tract or the stomach. If the lining epithelium is consistent with respiratory epithelium it is called bronchiogenic cyst. The present case is of an enterogenous cyst (bronchiogenic type) located at high cervical region, a rare site.
This 18 year old boy had radiating pain and progressive weakness of the right upper limb of six weeks duration. Neurological examination revealed grade 3 power in proximal group of muscles and grade 2 in distal muscles of right upper limb. There was hypertonia in all the four limbs with exaggerated deep tendon reflexes. His abdominal and cremasteric reflexes were absent and all other sensory modalities were well preserved. His biochemical and haematological parameters were within normal limits. MR imaging of cervical spine revealed an intradural extramedullary cystic lesion opposite C2/C3 bodies with signal intensities similar to that of CSF on both T1 and T2 weighted images [Figure.1]. C2-C4 laminectomy was done and an intradural extramedullary thin walled cyst with clear fluid, located ventrolateral to the cord on right side, was excised in toto. Three months later he had no neurological deficit. Histopathological examination of the excised tissue revealed cyst wall with papilleferous projections lined at some places by ciliated columnar epithelium with cilia and without cilia and at other places by pseudo stratified ciliated columnar epithelium. No ependymal or glial tissue was seen. Histopathology was consistent with bronchiogenic cyst [Figure. 2].
Enterogenous cyst is formed due to development anomaly. It is an endodermal remnant seen at abnormal site. These cysts result from persistence of abnormal communication between endodermal and neurectodermal tissue through a mesodermal layer defect. It is often associated with spinal abnormalities particularly of the vertebral body. Endodermal diverticulum may persist completely or a part of it may persist resulting in an isolated cyst located ventral to the cord with no vertebral body anomaly. Such cysts have been reported to occur from posterior fossa to lumbosacral region.[1] These lesions typically occur in dorsal spine or cervico-dorsal junction as cephalic end of the notochord develops first and is most sensitive to disturbance at that time.[2],[3] It is considered rare to find enterogenous cyst above C3 level. There were only nine cases of enterogenous cysts reported in high cervical region and only one case was reported from India.[4] There was one report where double intra spinal enterogenous cysts were present.[5]
Enterogenous cysts, histopathologically, can mimic any of the structures that originate from primitive foregut like respiratory system, oesophagus or stomach. Bronchiogenic cysts are formed of tissue, proper to respiratory system.[6],[7] Wilkin and Odoni[8] classified all types of enterogenous cysts into three groups depending on histopathological features, particularly depending on the presence of other tissue elements. Group A (simplest type) enterogenous cyst lined by a layer of cuboidal or columnar epithelium with or without cilia; Group B cyst with additional elements found along the course of gastro intestinal tract or tracheo-bronchial tree e.g. mucous glands and smooth muscle; and Group C cyst with ependymal or glial tissue in addition to foregut tissue. The present case had a cyst located anterior to cord at C2 level lined by ciliated columnar and pseudostratified ciliated epithelium with no other tissue element. It is not associated with any vertebral body anomaly. It is a group A enterogenous cyst of bronchiogenic type.[4]
There are only few reported cases of enterogenous cysts. Bronchiogenic cysts are still rare. Only nine cases have been reported so far; and only one case was reported in detail.[2],[3],[6],[7],[8],[9],[10],[11] These cases require surgical intervention. Total excision of the cyst may or may not be possible. The results with partial excision are as good as total excision. Overall prognosis is excellent in this condition.
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2. | Angoli AL, Laun A, Schonmayr R : Enterogenous intraspinal cysts. J Neurosurg 1984; 61 : 834-840. |
3. | Fabini GCA, Adams JE : High cervical spinal cord compression by an enterogenous cyst : Case report. J Neurosurg 1979; 51 : 556-559. |
4. | Bannur U, Rajekar V, Chandy MJ : High cervical intraspinal enterogenous cyst. Neurol India 1997; 45 : 98-100. |
5. | Chen IH, Kao KP, Penn W, HO DM : Double intraspinal enterogenous cysts. J Neurol Neurosurg Psychiatry 1995; 58 : 110. |
6. | Yamashita J, Anthony FJ Maloney, Harris P : Intradural spinal bronchiogenic cyst : case report. J Neuro Surg 1973; 39 : 240-245. |
7. | Mahanty S, Rao CJ, Shukla PK et al : Intradural enterogenous cyst. J Neurol Neurosurg Psychiatry 1979; 42 : 419-421. |
8. | Wilkins RH, Odoni GL : Spinal intradural cysts In : Hand Book of Clinical Neurology, Vinken PJ, Bruyn GW (Eds.) Vol.20, North Holland Publishers Amsterdam 1976; 55-102. |
9. | Arai Y, Yamauchi Y, Takaaki T Suji et al : Spinal Neurentic cyst : Report of two cases and review of forty one cases reported in Japan. Spine 1992; 17 : 1421-24. |
10. | Deshpande DH, Pandya SK, Dastur HM et al : Intraspinal enterogenous cyst. Neurol India 1972; 20 : 217-220. |
11. | Osenbach RK, Godersky JC, Trayneils et al : Intradural extramedullary cysts of spinal canal : Clinical presentation, radio graphic diagnosis and surgical management. Neurosurg 1992; 30 : 35-42. |
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