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Table of Contents    
LETTER TO EDITOR
Year : 2016  |  Volume : 64  |  Issue : 5  |  Page : 1083-1084

Bronchogenic intraspinal cyst – A rare case of spinal cystic space occupying lesion


1 Department of Neurosurgery, KMC, Manipal University, Manipal, Karnataka, India
2 Department of Neurosurgery, Bombay Hospital and Medical Research Centre, Mumbai, Maharashtra, India

Date of Web Publication12-Sep-2016

Correspondence Address:
Rajesh P Nair
Department of Neurosurgery, KMC, Manipal University, Manipal, Karnataka
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0028-3886.190262

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How to cite this article:
Vinod K, Nair RP, Deopujari C K. Bronchogenic intraspinal cyst – A rare case of spinal cystic space occupying lesion. Neurol India 2016;64:1083-4

How to cite this URL:
Vinod K, Nair RP, Deopujari C K. Bronchogenic intraspinal cyst – A rare case of spinal cystic space occupying lesion. Neurol India [serial online] 2016 [cited 2019 Nov 17];64:1083-4. Available from: http://www.neurologyindia.com/text.asp?2016/64/5/1083/190262




A 45-year-old male patient presented with progressive back pain of 2 month duration, aggravated on lying down, associated with weakness and numbness in the lower limbs since 1 month. On neurological examination, the tone was increased in both the lower limbs with a subjective power of grade 4/5 in the left lower limb and 4+/5 in the right lower limb. Ankle and knee jerks were brisk with hypoaesthesia below bilateral L1 dermatomes. Plantars were upgoing. A clinical diagnosis of a D10 spinal level, extramedullary lesion was made. Magnetic resonance imaging (MRI) of the dorsolumbar spine showed an intradural, extramedullary ventrally placed lesion at the D11–D12 level with significant compression of the spinal cord [Figure 1]. The lesion appeared hyperintense on T2W image with contrast enhancement and with significant compression and shift of the cord to the left [Figure 2]. After preoperative routine hematological investigations, he was taken for a D11–D12 laminectomy and total excision of the cystic lesion was performed [Figure 3]. Histological examination revealed ciliated columnar cells with areas of squamous metaplasia, and a pathological diagnosis of bronchogenic cyst was rendered [Figure 4]. The patient had an uneventful postoperative period with complete relief of symptoms at 3 week and 3 month follow-up.
Figure 1: Preoperative magnetic resonance imaging (dorsolumbar spine) sagittal sections, showing a hypertintense T2W and contrast enhancing ventral lesion at D11–D12 junction with significant cord compression ventrally

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Figure 2: Axial DL spine magnetic resonance imaging showing the T2W hyperintense lesion involving the entire spinal canal at the level of D11 and significant compression of the cord and shift to the left at the level of D12. Intraoperative ultrasonography used to identify the cystic intradural lesion at D11–D12

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Figure 3: Postoperative magnetic resonance imaging D-L spine (saggital sections) revealing gross total excision of the tumor with postoperative changes on T2W and contrast images

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Figure 4: Histological examination revealed ciliated columnar cells with areas of squamous metaplasia suggestive of an intraspinal bronchogenic cyst

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Intraspinal bronchogenic cysts are rare, benign, congenital cystic lesions. They arise as developmental abnormalities of the embryonic foregut.[1],[2],[3],[4] They are usually formed as a result of an accessory lung bud becoming isolated from rest of the tracheo-bronchial tree.[5] The lesion is usually in the form of a solitary benign, extrapulmonary cyst, found most commonly in the mediastinum with rare occurrence in the skin or subcutaneous tissues. It is poorly recognized by clinicians, and in almost all cases, the diagnosis is established by histopathology.

These cysts have been reported to occur from the posterior fossa to the lumbosacral region. Lesions typically occur in the dorsal spine or the cervicodorsal junction as the cephalic end of the notochord develops first and is most sensitive to disturbances occurring at that time. It is considered rare to find a bronchogenic cyst above the C3 level.

The cysts are typically intradural extramedullary lesions, most commonly located at the level of the cervical and thoracolumbar spine. They usually have a ventral location and are associated with other malformations of the spine such as fused vertebrae, spina bifida, and hemivertebrae.[6]

Various embryological hypotheses have been postulated, which include the (a) ectoedodermal adhesion theory,[7] (b) the cyst originating from the ectoderm, which is capable of producing both endodermal and mesodermal tissues,[8] and (c) a third theory called the “split notochord syndrome” postulated by Bentley and Smith that describes an incomplete duplication and separation of the notochord.[2],[9]

Wilkins and Odoms [10] suggested three histological categories based on microscopic features

  • Category A–simple cyst lined by epithelium on a basement membrane with a thin wall of connective tissue
  • Category B–cyst lined by epithelium with a wall containing tissues found along the gastrointestinal tract or tracheobronchial tree
  • Category C–cyst lined by epithelium with a wall containing ependymal and glial tissues as an intrinsic part of the lesion.


In our case, we describe an intradural, extramedullary bronchogenic cyst (Wilkins and Odoms Category B), located ventrally in the dorsal canal with no associated vertebral anomalies.

These lesions grow slowly owing to tight junctions between the epithelial cells, limiting the expansion of the cyst.[11],[12] The most effective treatment of a symptomatic spinal bronchogenic cyst is surgical resection. However, complete resection can be challenging if the cyst is adherent to the spinal cord.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
  References Top

1.
Rao GP, Bhaskar G, Reddy PK. Cervical intradural extramedullary bronchogenic cyst. Neurol India 1999;47:79-81.  Back to cited text no. 1
[PUBMED]  Medknow Journal  
2.
Rauzzino MJ, Tubbs RS, Alexander E 3rd, Grabb PA, Oakes WJ. Spinal neurenteric cyst and their relation to more common aspects of occult spinal dysraphism. Neurosurg Focus 2001;10:e2.  Back to cited text no. 2
    
3.
Baumann CR, Könü D, Glatzel M, Seigel AM. Thoracolumbar intradural extramedullary bronchogenic cyst. Acta Neurochir (Wein) 2005;137:317-9.  Back to cited text no. 3
    
4.
Yamashita J, Maloney AF, Harris P. Intradural spinal bronchogenic cyst: Case report. J Neurosurg 1973;39:240-5.  Back to cited text no. 4
[PUBMED]    
5.
Sundaram C, Paul TR, Raju BV, Ramakrishna Murthy T, Sinha AK, Prasad VS, et al. Cysts of the central nervous system: A clinicopathologic study of 145 cases. Neurol India 2001;49:237-42.  Back to cited text no. 5
[PUBMED]  Medknow Journal  
6.
Jain SK, Chopra S, Mathur PP. Thoracic neurenteric cyst in a 60 year old male. Neurol India 2004;52:402-3.  Back to cited text no. 6
[PUBMED]  Medknow Journal  
7.
Macdonald RL, Schwartz ML, Lewis AJ. Neurenteric cyst located dorsal to the cervical spine: Case report. Neurosurgery 1991;28:583-7.  Back to cited text no. 7
[PUBMED]    
8.
Mann KS, Khosla VK, Gulati DR, Malik AK. Spinal neurenteric cyst. Association with vertebral anomalies, diastematomyelia, dorsal fistula, and lipoma. Surg Neurol 1984;21:358-62.  Back to cited text no. 8
[PUBMED]    
9.
Bentley JF, Smith JR. Developmental posterior enteric remnants and spinal malformations: The split notochord syndrome. Arch Dis Child 1960;35:76-86.  Back to cited text no. 9
[PUBMED]    
10.
Wilkins RH, Odom GL. Spinal intradural cysts. In: Vinken PJ, Bruyn GW, editors. Handbook of Clinical Neurology. Vol. 20. Amsterdam, Oxford, New York: North Holland Publishing 1976; pp. 55-102.  Back to cited text no. 10
    
11.
Ho KL, Tiel R. Intraspinal bronchogenic cyst: Ultrastructural study of the lining epithelium. Acta Neuropathol 1989;78:513-20.  Back to cited text no. 11
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12.
Ko KS, Jeun SS, Lee YS, Park CK. Sacral intraspinal bronchogenic cyst: A case report. J Korean Med Sci 2008;23:895-7.  Back to cited text no. 12
[PUBMED]    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]



 

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