Atormac
brintellex
Neurology India
menu-bar5 Open access journal indexed with Index Medicus
  Users online: 4454  
 Home | Login 
About Editorial board Articlesmenu-bullet NSI Publicationsmenu-bullet Search Instructions Online Submission Subscribe Videos Etcetera Contact
  Navigate Here 
 Search
 
  
 Resource Links
    Similar in PUBMED
   Search Pubmed for
   Search in Google Scholar for
    Article in PDF (1,324 KB)
    Citation Manager
    Access Statistics
    Reader Comments
    Email Alert *
    Add to My List *
* Registration required (free)  

 
  In this Article
   References
   Article Figures

 Article Access Statistics
    Viewed1035    
    Printed22    
    Emailed0    
    PDF Downloaded69    
    Comments [Add]    

Recommend this journal

 


 
Table of Contents    
LETTER TO EDITOR
Year : 2014  |  Volume : 62  |  Issue : 4  |  Page : 453-455

Suprasellar tuberculoma associated with unilateral moyamoya phenomenon: Case report


1 Department of Neurosurgery, National Institute of Mental Health and Neurosciences, Bangalore, Karnataka, India
2 Department of Neuropathology, National Institute of Mental Health and Neurosciences, Bangalore, Karnataka, India

Date of Web Publication19-Sep-2014

Correspondence Address:
Paritosh Pandey
Department of Neurosurgery, National Institute of Mental Health and Neurosciences, Bangalore, Karnataka
India
Paritosh Pandey
Department of Neurosurgery, National Institute of Mental Health and Neurosciences, Bangalore, Karnataka
India
Login to access the Email id

Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0028-3886.141264

Rights and Permissions



How to cite this article:
Pruthi N, Karanth S, Nagarjun M N, Pandey P, Pruthi N, Karanth S, Nagarjun M N, Pandey P. Suprasellar tuberculoma associated with unilateral moyamoya phenomenon: Case report . Neurol India 2014;62:453-5

How to cite this URL:
Pruthi N, Karanth S, Nagarjun M N, Pandey P, Pruthi N, Karanth S, Nagarjun M N, Pandey P. Suprasellar tuberculoma associated with unilateral moyamoya phenomenon: Case report . Neurol India [serial online] 2014 [cited 2020 Oct 31];62:453-5. Available from: https://www.neurologyindia.com/text.asp?2014/62/4/453/141264


Sir,

Suprasellar tuberculomas are rare and commonly present with visual disturbances or features of hypopituitarism. Some suprasellar tumors have been known to be associated with unilateral or bilateral occlusion of internal carotid artery along with moyamoya phenomenon. [1],[2] However, this phenomenon has not been described with suprasellar tuberculomas. We report one such case.

An 8-year-old boy presented with features of raised intracranial pressure and worsening of vision for the last 3 months. There was no other relevant history. On examination, the vision was 6/12 in the right eye and 6/24 in the left eye, with bitemporal hemianopia. Magnetic resonance imaging (MRI) showed a T1 isointense and T2 hypointense tumor with small cysts, extending to the floor of third ventricle, suggestive of a suprasellar tumor, probably a craniopharyngioma. In addition, there was complete non-visualization of left internal carotid artery (ICA), with small flow voids in the region of ICA bifurcation. Digital subtraction angiography (DSA) showed left ICA injection revealed complete occlusion of ICA distal to posterior communicating artery, and formation of some moyamoya collaterals. Bilateral anterior cerebral arteries (ACAs) were filling from the right A2. On ECA injection, the entire cerebral circulation was supplied by multiple dural collaterals, mainly from the middle meningeal artery. There was no stenosis or occlusion on right ICA injection [Figure 1]a-l.

Because the dural collaterals were supplying the brain on the left side, the patient was operated from the right side. A right pterional craniotomy was done, and the tumor was partially decompressed. Partial decompression of the lesion was done, and lamina terminalis was opened for hydrocephalus. Postoperatively, his vision improved to 6/6 in both eyes, and his visual fields normalized. Histopathology report showed an aggregate of lymphocytes, histiocytes, and plasma cells forming an ill-formed epithelioid cell granuloma with Langhans giant cells. AFB was negative. A provisional diagnosis of tuberculoma was made [Figure 2]. Considering the histopathology report, he was started on anti-tubercular treatment in weight-appropriate doses; at 3-month follow up, he was asymptomatic.
Figure 1: (a) CT Head contrast showing a calcified mass in the suprasellar region; (b-f) MRI, T1WI, T2W1, Coronal T2WI, T1WI contrast axial, and coronal, respectively showing a T1 hypo, T2 hypointense lesion with peripheral enhancement; (g and h) MR angiogram images showing complete occlusion of Left ICA; (i-l) DSA, Left ICA AP, and lateral projections showing complete occlusion of left ICA beyond ophthalmic artery with moyamoya collaterals; (c and d) Left ECA AP and lateral projections showing excellent revascularization of from ECA collaterals

Click here to view
Figure 2: An aggregate of lymphocytes, histiocytes, and plasma cells forming an ill-formed epithelioid cell granuloma (×20, H and E). Inset highlights the Langhans giant cell (×40)

Click here to view


Suprasellar tuberculomas are very rare lesions. Sharma et al. described a large series of sellar tuberculomas, which accounted for 1.5% of all sellar pathologies managed in their institution. [3] Other authors have described reports of suprasellar tuberculomas presenting with visual deficits, hypopituitarism, or a mass associated with tubercular infection elsewhere in the body. [4]

Moyamoya syndrome is rarely associated with pediatric suprasellar tumors. It is most common after radiation therapy of the suprasellar tumors. [5] The association of moyamoya syndrome with suprasellar tumors in absence of treatment is very rare. Tsuji et al. presented a rare association of craniopharyngioma with moyamoya syndrome. [6] There are other reports of moyamoya syndrome associated with craniopharyngiomas, and in one case, a growth hormone-secreting pituitary adenoma and germinoma. This is probably the first case of moyamoya syndrome associated with a suprasellar tuberculoma. There could have been an associated sub-clinical meningitic syndrome with the tuberculoma, which led to the development of ICA occlusion secondary to arteritis, or it may be an unusual association of 2 diseases. The ICA occlusion and moyamoya syndrome was probably very long-standing because there was complete revascularization of the entire hemisphere from collaterals from external carotid artery branches, particularly MMA. In the present patient, there was excellent natural revascularization, and hence, no need for treatment of the moyamoya syndrome.

In conclusion, the moyamoya syndrome associated with suprasellar tumors is very rare, and this is the first report of its association with suprasellar tuberculoma. It is important to diagnose the vascular occlusion and the extent of revascularization preoperatively to avoid any postoperative ischemia.

 
  References Top

1.Arita K, Uozumi T, Oki S, Kuwabara S, Ohba S, Nakahara T, et al. Moyamoya disease associated with pituitary adenoma-report of two cases. Neurol Med Chir (Tokyo) 1992;32:753-7.  Back to cited text no. 1
    
2.Lau YL, Milligan DW. Atypical presentation of craniopharyngioma associated with Moyamoya disease. J R Soc Med 1986;79:236-7.  Back to cited text no. 2
    
3.Sharma MC, Arora R, Mahapatra AK, Sarat-Chandra P, Gaikwad SB, Sarkar C. Intrasellar tuberculoma-an enigmatic pituitary infection: A series of 18 cases. Clin Neurol Neurosurg 2000;102:72-7.  Back to cited text no. 3
    
4.Nayil K, Singh S, Makhdoomi R, Ramzan A, Wani A. Sellar-suprasellar tuberculomas in children: 2 cases and literature review. Pediatr Neurol 2011;44:463-6.  Back to cited text no. 4
    
5.Lee HS, Seol HJ, Kong DS, Shin HJ. Moyamoya syndrome precipitated by cranial irradiation for craniopharyngioma in children. J Korean Neurosurg Soc 2011;50:535-7.  Back to cited text no. 5
    
6.Tsuji N, Kuriyama T, Iwamoto M, Shizuki K. Moyamoya disease associated with craniopharyngioma. Surg Neurol 1984;21:588-92.  Back to cited text no. 6
    


    Figures

  [Figure 1], [Figure 2]



 

Top
Print this article  Email this article
   
Online since 20th March '04
Published by Wolters Kluwer - Medknow