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Table of Contents    
LETTER TO EDITOR
Year : 2012  |  Volume : 60  |  Issue : 6  |  Page : 665-667

Combined pituitary adenoma and Rathke's cleft cysts: Two multicystic cases and literature review


Department of Neurosurgery, West China Hospital, Sichuan University, Chengdu, Sichuan Province, P.R., China

Date of Submission05-Sep-2012
Date of Decision05-Sep-2012
Date of Acceptance05-Sep-2012
Date of Web Publication29-Dec-2012

Correspondence Address:
Shu Jiang
Department of Neurosurgery, West China Hospital, Sichuan University, Chengdu, Sichuan Province, P.R.
China
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0028-3886.105217

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How to cite this article:
Zhou P, Cai B, Ma W, Jiang S. Combined pituitary adenoma and Rathke's cleft cysts: Two multicystic cases and literature review . Neurol India 2012;60:665-7

How to cite this URL:
Zhou P, Cai B, Ma W, Jiang S. Combined pituitary adenoma and Rathke's cleft cysts: Two multicystic cases and literature review . Neurol India [serial online] 2012 [cited 2019 Oct 21];60:665-7. Available from: http://www.neurologyindia.com/text.asp?2012/60/6/665/105217


Sir,

Coexistence of pituitary adenoma and Rathke's cleft cyst (RCC) are relatively uncommon, and lesions with multicystic presentation are extremely rare. [1],[2],[3],[4],[5],[6]

A 38-year-old woman was referred with headache, exophthalmus, and Cushing's syndrome. She was a diagnosed case of pituitary adrenocorticotropic hormone (ACTH) secreting adenoma. The basal ACTH value was 188.9 ng/L and free cortisol value of 24-hour urine was 939.3 ug. The invasive lesion contained two different signal intensities on magnetic resonance imaging (MRI) [Figure 1]a-c. The main lesion showed iso-signal intensity on T1-and T2-weighted imaging, the micro-cysts embedded in the iso-signal lesion showed hybrid signal on T2-weighted images and "slit" like low-signal intensity on T1-weighted images. The lesion was resected subtotally via a transsphenoidal surgery and no obvious fluid was detected. Histology disclosed multicystic RCCs that were confirmed by the cytokeratin positive ciliated columnar cells embedded in the corticotroph adenoma elements [Figure 1]d. Postoperative course was uneventful. Gamma knife surgery was opted for the residual tumor three month after the surgery.
Figure 1: Preoperative MRI scan (a,b,c) and histological findings (d) of case 1. Sagittal T1-weighted image (a) and axial T2-weighted image (b) show an intrasellar mass with two different signal intensities, the "slit" like low-signal intensity cysts can also detected (a) Post-contrast image showed marked enhancement of the solid part and the non-enhanced cysts (c) Multiple mucin-filled cysts are interspersed within the adenoma
tissue (d, H and E, X200)


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A 38-year-old woman was referred with visual disturbance of 12 months duration. He underwent transcranial surgery for the partial resection of a non-functioning pituitary adenoma 6 months before his administration. Biochemical investigations showed hypopituitarism. MRI showed an invasive lesion with iso-signal intensity mass combined with diffused high-signal intensity elements on T1-weighted images and enhanced adenoma [Figure 2]a and c. On T2-weighted images, the lesion showed some high-signal intensity elements impacted in the iso-signal intensity mass [Figure 2]b. The residual tumor was subtotally resected via transcranial surgery. Postoperative cerebrospinal fluid rhinorrhea subsided after the lumbar drainage combined with transsphenoidal sellar floor repairation. No other operative complications occurred and her visual acuity recovered. Histology revealed multilocular RCCs and cholesterin were embedded in the pituitary adenoma lesion [Figure 2]d-f, the adenoma cells were positive for CgA [Figure 2]d and e by immunohistochemical staining and the Ki-67 index was less than 1%.
Figure 2: Preoperative MRI scan (a, b, c) and histological findings (d) of case 2. Multilocular RCCs (e,f) and cholesterin (d) were embedded in the pituitary adenoma lesion (d, e, f), the adenoma cells were positive for CgA

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The coexistence of RCC and pituitary adenoma is very rare. In the large series reported, RCCs were associated with 0.51-1.7% of pituitary adenomas. [1],[2],[3],[4],[5] In our database, five such cases (0.62%) were identified in 801 patients with a confirmed pituitary adenoma. Only two cases with multicysts were identified associated with pituitary adenoma. The diagnosis of combined pituitary adenoma and RCC is rarely made before surgery. RCCs show variable position, size, and signal intensity on MRI, which depends on protein concentration. RCCs are rarely enclosed in pituitary adenomas and are frequently adjacent to the adenomas. Most authors thought that the occurrence of non-enhancing cyst on the MRI evaluation for pituitary adenoma may be helpful for such coexistence lesions. [6] Based on the MRI and histological findings of our two patients, unlike the heterogeneous signal intensity cyst on MRI, the identification of homogeneous micro cysts within the solid tumor may be a sign of RCCs. Histological examination seems more important than the radiographic and clinical findings. Most RCCs have ciliated epithelial cells that are positive for cytokeratin, but the hemorrhagic lesions have no such elements. Although the RCC and pituitary adenoma have the same ancestry, the relationship between pituitary adenomas and RCCs is uncertain. Associations between these lesions have been considered as purely coincidental. Recently, many unusuals were reported, such as combined gangliocytoma and non-functioning pituitary adenoma, the presence of intermediate cells, ependymal cells, and nerve bundle structure within pituitary adenoma. As the progenitor cells were identified in the adult pituitary, pituitary adenoma stem cell-like cells were also isolated. We thought that the RCCs may be derived from the stimulation of stem cell-like cells of adenoma tissue, as happened in our two multicystic RCCs within the adenoma.


 » Acknowledgment Top


These authors were supported by the National Natural Science Foundation of China (Grant No. 81072077) and Foundation of Science and Technology Department of Sichuan Provience (Grant NO.2010SZ0160).

 
 » References Top

1.Noh SJ, Ahn JY, Lee KS, Kim SH. Pituitary adenoma and concomitant Rathke's cleft cyst. Acta Neurochir (Wien) 2007;149:1223-8.  Back to cited text no. 1
[PUBMED]    
2.Teramoto A, Hirakawa K, Sanno N, Osamura Y. Incidental pituitary lesions in 1,000 unselected autopsy specimens. Radiology 1994;193:161-4.  Back to cited text no. 2
[PUBMED]    
3.Nishio S, Mizuno J, Barrow DL, Takei Y, Tindall GT. Pituitary tumours composed of adenohypophysial adenoma and Rathke's cleft cyst elements. A clinicopathology study. Neurosurgery 1987;21:371-7.  Back to cited text no. 3
[PUBMED]    
4.You C, Qiao F, Jiang S, Xiao A. Growth hormone secreting pituitary adenoma associated with Rathke's cleft cyst. Neurol India 2012;60:310-1.  Back to cited text no. 4
[PUBMED]  Medknow Journal  
5.Wang K, Ma L, You C. Pituitary adenoma and concomitant Rathke's cleft cyst: A case report and review of the literature. Neurol India 2012;60:309-10.  Back to cited text no. 5
[PUBMED]  Medknow Journal  
6.Sumida M, Migita K, Tominaga A, Iida K, Kurisu K. Concomitant pituitary adenoma and Rathke's cleft cyst. Neuroradiology 2001;43:755-9.  Back to cited text no. 6
[PUBMED]    


    Figures

  [Figure 1], [Figure 2]

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