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LETTER
Year : 2014  |  Volume : 60  |  Issue : 4  |  Page : 421-422

Lymphocytic hypophysitis in non-pregnant middle-aged females mimicking pituitary adenoma: A report of two cases


1 Department of Pathology, Bhopal Memorial Hospital and Research Centre (BMHRC), Bhopal, Madhya Pradesh, India
2 Department of Neurosurgery, Bhopal Memorial Hospital and Research Centre (BMHRC), Bhopal, Madhya Pradesh, India

Date of Web Publication5-Nov-2014

Correspondence Address:
Dr. M Tripathi
Department of Pathology, Bhopal Memorial Hospital and Research Centre (BMHRC), Bhopal, Madhya Pradesh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0022-3859.144000

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How to cite this article:
Gulwani H, Tripathi M, Garg N. Lymphocytic hypophysitis in non-pregnant middle-aged females mimicking pituitary adenoma: A report of two cases. J Postgrad Med 2014;60:421-2

How to cite this URL:
Gulwani H, Tripathi M, Garg N. Lymphocytic hypophysitis in non-pregnant middle-aged females mimicking pituitary adenoma: A report of two cases. J Postgrad Med [serial online] 2014 [cited 2019 Oct 14];60:421-2. Available from: http://www.jpgmonline.com/text.asp?2014/60/4/421/144000


Sir,

Lymphocytic hypophysitis (LH) is an autoimmune disease characterized by infiltration of the pituitary gland by inflammatory cells that result in pituitary expansion and a variable degree of hypopitutarism. It closely mimics features of pituitary adenoma clinically and sometimes radiologically. [1] LH most commonly affects pregnant and peripartum females [2] who usually present with headache and visual disturbances. However, LH can be encountered in non-pregnant females and males as well. Herein we describe two such unusual cases of LH that mimicked clinically as pituitary adenomas.

The first case was a 40-year-old woman who presented with history of headache, diplopia and sixth cranial nerve paresis [Figure 1]a-f. Her hormonal profile was within normal limits except for mild hyperprolactinemia. The second case was that of a 35-year-old lady who presented with history of headache of a month's duration that was holocranial and moderate in intensity [Figure 2]a-c. Her hormonal profile was within normal limits. MRI of both cases had similar appearance and revealed a sellar lesion with suprasellar extension causing indentation on optic chiasma. There was uniform enhancement along the pituitary stalk and dura along the adjoining skull base. Both patients underwent surgery with partial excision of the lesion due to its firm consistency. Diagnosis of LH was made based on histological examination. Hematoxylin and eosin (H&E)-stained sections from the first case revealed distortion of the pituitary gland by heavy lymphoplasmacytic inflammatory cell infiltrate. The pituitary gland in surrounding area showed few intact acini that were delineated by reticulin fibers. Toluidine blue stain demonstrated mast cells in vicinity of capillaries and other areas. Sections from the second case also showed similar destruction of pituitary gland by lymphomononuclear cells. Focal atrophy and prominent oncocytic change was noted in some acini. Post-operatively, the first patient responded well with recovery of 6 th cranial nerve deficit but developed hypothyroidism and hypocortisolism for which she was maintained on hormone replacement therapy. Her follow-up MRI showed no evidence of recurrence with adequately decompressed optic chiasma. Postoperative period was uneventful for the second patient.
Figure 1: (a) MRI T1W sagittal with lesion in sella. (b) T1W with contrast enhancement of lesion and adjacent dura. (c) T1W coronal with contrast lesion extending up to optic chiasma. (d) T1W with contrast (Follow up) with residual enhancement of pituitary stalk. (e) Photomicrograph showing destruction of pituitary gland by lymphoplasmacytic cells (H&E, x40). (f) Toluidine blue showing increased mast cells. (Toluidine blue stain, x40)

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Figure 2: (a) MRI T1W with contrast with enhancement of lesion and associated dura along upper clivus. (b) Pituitary stalk enhancement. (c) Photomicrograph showing destruction of pituitary gland by lymphoplasmacytic cells and oncocytic change of residual pituitary acini on right. (H&E, x40)

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LH closely mimics pituitary adenoma but can be differentiated due to predilection for destruction of ACTH- and TSH-secreting cells in former. In pituitary adenoma ACTH-secreting cells are last to be affected. [3] Non-functioning adenomas also present with mass effects or hormone deficiencies but hypogonadism rather than hypocortisolism and diabetes insipidus is usually present in them. [1] Therefore, early detection and accurate diagnosis by neuroimaging and histopathology is of vital importance for proper management of LH patients. MRI scan in LH patients shows symmetrical pituitary enlargement with extra sellar extension which usually displaces optic chiasma. Pitutary stalk is usually not deviated. [4] Gadolinium scan in these patients reveals intense homogenous enhancement of the lesion along with that of the pituitary stalk and dural tail. [2] However, pituitary adenoma patients have asymmetrical pituitary enlargement with deviated stalk and on gadolinium scan enhancement without dural tail is noted. [2] In conclusion, LH is not uncommon in non-pregnant middle-aged females, so a possibility of LH should always be kept in mind when such patients present with non-specific symptoms and atypical features on neuroimaging. Surgery should be limited to biopsy or to decompress optic chiasma as it can cause deleterious effect on endocrinal functions. Patients can develop hypopituitarism for which they have to take lifelong hormone replacement therapy.

 
 :: References Top

1.
Thodou E, Asa SL, Kontogeorgos G, Kovacs K, Horvath E, Ezzat S. Clinical case seminar: Lymphocytic hypophysitis: Clinicopathological findings. J Clin Endocrinol Metab 1995;80:2302-11.  Back to cited text no. 1
    
2.
Bellastella A, Bizzarro A, Coronella C, Bellastella G, Sinisi AA, De Bellis A. Lymphocytic hypophysitis: A rare or underestimated disease? Eur J Endocrinol 2003;149:363-76.  Back to cited text no. 2
    
3.
Cuthbertson DJ, Ritchie D, Crooks D, Main G, Smith C, Vora J, et al. Lymphocytic hypophysitis occurring simultaneously with a functioning pituitary adenoma. Endocr J 2008;55:729-35.  Back to cited text no. 3
    
4.
Abe T, Matsumoto K, Sanno N, Osamura Y. Lymphocytic hypophysitis: Case report. Neurosurgery 1995;36:1016-9.  Back to cited text no. 4
    


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