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TOPIC OF THE ISSUE: LETTER TO EDITOR
Year : 2012  |  Volume : 60  |  Issue : 3  |  Page : 307-308

Pituitary adenoma presenting with Cerebrospinal fluid rhinorrhea as the sole symptom


Department of Neurosurgery, All India Institute of Medical Sciences, New Delhi-110029, India

Date of Submission02-Aug-2011
Date of Decision03-Aug-2011
Date of Acceptance19-Aug-2011
Date of Web Publication14-Jul-2012

Correspondence Address:
Ashok K Mahapatra
Department of Neurosurgery, All India Institute of Medical Sciences, New Delhi-110029
India
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Source of Support: None, Conflict of Interest: None


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How to cite this article:
Goyal N, Borkar SA, Agrawal D, Mahapatra AK. Pituitary adenoma presenting with Cerebrospinal fluid rhinorrhea as the sole symptom. Neurol India 2012;60:307-8

How to cite this URL:
Goyal N, Borkar SA, Agrawal D, Mahapatra AK. Pituitary adenoma presenting with Cerebrospinal fluid rhinorrhea as the sole symptom. Neurol India [serial online] 2012 [cited 2021 Dec 3];60:307-8. Available from: https://www.neurologyindia.com/text.asp?2012/60/3/307/98519


Sir,

Cerebrospinal fluid (CSF) rhinorrhea is a known complication of trans-sphenoidal surgery, radiotherapy, or bromocriptine therapy [1],[2] but is an extremely rare presentation of untreated pituitary adenoma [Table 1]. [3],[4],[5],[6],[7],[8],[9],[10],[11],[12],[13] This report presents one such case.
Table 1: Summary of reported cases of untreated pituitary adenoma presenting with CSF rhinorrhea

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A 35-year-old man presented with a history of watery discharge through the nose of 6-month duration. On examination, visual acuity was 6/9 in both the eyes with no visual field defect. His hormone profile and CSF analysis was normal. Contrast computed tomography (CT) of head and contrast magnetic resonance imaging (MRI) of brain revealed a pituitary macroadenoma eroding sellar floor [Figure 1] and [Figure 2]. He subsequently underwent tumor decompression by transnasal trans-sphenoidal approach. Tumor was soft in consistency and moderately vascular. Normal pituitary could be identified separately. A small defect was identified in the arachnoid and dura, through which CSF was leaking. The defect was packed with fat, fascia, and tissue glue. Lumbar CSF drain was placed in the postoperative period, which was removed on the fifth postoperative day. There was no CSF rhinorrhea postoperatively. Histopathology report confirmed the diagnosis of pituitary adenoma, which was immunonegative for follicle stimulating hormone (FSH), luteinizing hormone (LH), growth hormone (GH), thyroid stimulating hormone (TSH), adrenocorticotropic hormone (ACTH) and prolactin.
Figure 1: Magnetic resonance imaging of the brain with contrast: axial (a), sagittal (b), and coronal (c) views showing a pituitary macroadenoma with extension into the sphenoid sinus. It can be seen that that there is minimal supradiaphragmatic extension

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Figure 2: Computed tomography of the head with bone windows: sagittal (a) and coronal (b) views showing direct erosion of the sellar floor by the tumor

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CSF rhinorrhea can be broadly classified into traumatic and nontraumatic; the former comprising the vast majority of cases. When compared to traumatic CSF rhinorrhea, the rarer but more serious nontraumatic type has a lower incidence of spontaneous closure. [14] The pathogenesis of CSF rhinorrhea after trans-sphenoidal surgery is not difficult to understand. The hypothesis for the occurrence of CSF rhinorrhea following medical therapy or radiation is that these therapies lead to shrinkage of the tumor, which may be plugging the defect thereby preventing the leak and shrinkage of the tumor may result in CSF leakage. [1],[2] Ommaya et al. [14] classified nontraumatic CSF leakage caused by tumors into two types: direct and indirect, based on whether the tumor directly erodes the meninges and bone, or indirectly causes erosion of anatomically fragile areas of the skull base, as a result of the increased intracranial pressure. The mechanism of CSF rhinorrhea in patients with untreated pituitary adenoma is not well understood and a number of hypotheses have been postulated. Most authors cite erosion of the sellar floor by the tumor through the skull base as the cause. Nutkiewicz et al. [5] hypothesized that combination of a strong and competent diaphragma sellae and a fold of evaginated arachnoid membrane may force pituitary tumors to expand inferiorly, rather than superiorly, thereby favoring CSF leakage into the sphenoid sinus. According to Fager et al., [1] the tumor may function as a "stopper," which sometimes undergoes spontaneous necrosis as a result of hemorrhage or infarction, leading to unblocking the stop in the CSF flow through the defect. [1] In females, spontaneous regression of the tumor may also occur as a result of decreased estrogen level at menopause. [10] According to Obana et al., [8] the tumor causes intracranial hypertension, which is relieved by naturally shunting the CSF through an anatomically fragile area in the base of the skull. [8] In this patient, direct erosion of the sellar floor by the tumor, as visualized on neuroimaging, is the most likely cause of CSF rhinorrhea.

A high index of suspicion is required to diagnose pituitary adenoma when the presenting feature in these patients is CSF rhinorrhea, as CSF rhinorrhea in these patients can be misdiagnosed as allergic rhinitis. [15] Ahistory of associated visual and endocrinal disturbances is helpful. However, rarely pituitary adenoma can present solely with rhinorrhea, as observed in our patients. A careful inquiry regarding history of salty fluid trickling down the throat should be made. If the watery discharge is copious and can be collected, it should be analyzed for CSF. [15] A CT cisternography or an MRI scan with CISS sequence is warranted as tumors of sellar region can cause spontaneous rhinorrhea. The definitive management includes removal of the tumor and packing of the defect. [8],[11]

 
  References Top

1.Fager CA. Nature and treatment of cerebrospinal fluid rhinorrhea in pituitary tumors. Surg Clin North Am 1973;53:283-90.  Back to cited text no. 1
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2.Barlas O, Bayindir Ç, Hepgül K, Can M, Kiriº T, Sencer E, et al . Bromocriptine-induced cerebrospinal fluid fistula in patients with macroprolactinomas: Report of three cases and a review of the literature. Surg Neurol 1994;41:486-9.  Back to cited text no. 2
    
3.Giovanelli M, Perria C. Cerebrospinal rhinorrhea with pituitary adenoma. (Case report). Acta Neurochir (Wien) 1967;16:261-6.  Back to cited text no. 3
[PUBMED]    
4.Cole IE, Keene M. Cerebrospinal fluid rhinorrhea in pituitary tumors. J R Soc Med 1980;73:244-254.  Back to cited text no. 4
[PUBMED]  [FULLTEXT]  
5.Nutkiewicz A, DeFeo DR, Kohut RI, Fierstein S. Cerebrospinal fluid rhinorrhea as a presentation of pituitary adenoma. Neurosurgery 1980;6:195-7.  Back to cited text no. 5
[PUBMED]    
6.Rothrock JF, Laguna JF, Raynolds AF. CSF rhinorrhea from untreated pituitary adenoma. Arch Neurol 1982;39:442-3.  Back to cited text no. 6
    
7.Bilo HJ, Ponssen H, van der Veen EA, Wolbers JG. Rhinorrhea as the presenting symptom of pituitary adenoma. Clin Neurol Neurosurg 1984;86:47-9.  Back to cited text no. 7
[PUBMED]  [FULLTEXT]  
8.Obana WG, Hodes JE, Weinstein PR, Wilson CB. Cerebrospinal fluid rhinorrhea in patients with untreated pituitary adenoma: Report of two cases. Surg Neurol 1990;33:336-40.  Back to cited text no. 8
[PUBMED]    
9.Carroll WR, Cohen S, Sullivan MJ. Spontaneous CSF rhinorrhea: An unusual presentation of a pituitary adenoma. Otolaryngol Head Neck Surg 1991;104:380-3.  Back to cited text no. 9
[PUBMED]    
10.Ohtakara K, Matsubara T, Kojima T, Taki W, Waga S. Cerebrospinal fluid rhinorrhea associated with untreated prolactinoma: Case Report. Neurol Med Chir (Tokyo) 2000;40:413-8.  Back to cited text no. 10
[PUBMED]  [FULLTEXT]  
11.Hanel RA, Prevedello DM, Correa A, Antoniuk A, Araújo JC. Cerebrospinal fluid fistula as the presenting manifestation of pituitary adenoma: Case report with a 4-year follow-up. Arq Neuropsiquiatr 2001;59:263-5.  Back to cited text no. 11
    
12.Telera S, Conte A, Cristalli G, Occhipinti E, Pompili A. Spontaneous cerebrospinal fluid rhinorrhea as the presenting symptom of sellar pathologies: Three demonstrative cases. Neurosurg Rev 2007;30:78-82.  Back to cited text no. 12
[PUBMED]  [FULLTEXT]  
13.Makin V, Hatipoglu B, Hamrahian AH, Arrossi AV, Knott PD, Lee JH, et al. Spontaneous cerebrospinal fluid rhinorrhea as the initial presentation of growth hormone-secreting pituitary adenoma. Am J Otolaryngol 2011;32:433-7.  Back to cited text no. 13
[PUBMED]  [FULLTEXT]  
14.Ommaya AK. Cerebrospinal fluid fistula and pneumocephalus. In: Wilkins R, Rengachary S, editors. Neurosurgery. Vol 2, 2 nd ed. New York: McGraw Hill; 1996. p. 2773-82.  Back to cited text no. 14
    
15.Mohanty S, Tandon PN. Transcranial repair of CSF Rhinorrhea. In: Ramamurthi R, Sridhar K, Vasudevan MC, editors. Textbook of Operative Neurosurgery. vol 1, 1 st ed. New Delhi: BI Publications Pvt Ltd; 2005. p. 255-61.  Back to cited text no. 15
    


    Figures

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    Tables

  [Table 1]

This article has been cited by
1 Subarachnoid hemorrhage after transsphenoidal surgery for pituitary adenoma: A case report and review of literature
Goyal, N. and Basheer, N. and Suri, A. and Mahapatra, A.K.
Neurology India. 2012; 60(3): 337-338
[Pubmed]



 

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