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| LETTER TO EDITOR |
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| Year : 2005 | Volume
: 53
| Issue : 3 | Page : 363-364 |
Lymphocytic hypophysitis presenting as pituitary apoplexy in a male
B Minakshi, S Alok, KP Hillol
Departments of Pathology and Neurosurgery, Dr. Ram Manohar Lohia Hospital, New Delhi, India
| Date of Acceptance | 24-Feb-2005 |
Correspondence Address:
S Alok
Departments of Pathology and Neurosurgery, Dr. Ram Manohar Lohia Hospital, New Delhi
India
 Source of Support: None, Conflict of Interest: None  | Check |
DOI: 10.4103/0028-3886.16948
How to cite this article:
Minakshi B, Alok S, Hillol K P. Lymphocytic hypophysitis presenting as pituitary apoplexy in a male. Neurol India 2005;53:363-4 |
Sir,
Inflammatory lesions of the pituitary are rare with an estimated incidence of one case in ten million population.[1] Lymphocytic or autoimmune hypophysitis was first described by Goudie and Pickerton[2] in 1962, and since then approximately 379 cases have been reported.[3] Most cases occur in women, mostly during late pregnancy or early postpartum period. Pituitary apoplexy, a clinical syndrome of sudden onset of severe neurological dysfunction due to hemorrhage or infarction of pituitary, usually occurs in the setting of a pre-existing adenoma. Association of pituitary apoplexy and lymphocytic hypophysitis has been reported only twice earlier, both in female patients.[4],[5]
A 42-year male presented with progressive deterioration of vision in both eyes over a 1-year period. A contrast enhanced CT scan revealed the presence of a sellar mass suggestive of an adenoma. The patient was advised surgery, but he refused to accept this advice. He was brought in an unconscious state to our institution. A magnetic resonance (MR) image showed a sellar mass with suprasellar extension [Figure - 1], [Figure - 2]. He was treated managed with high-dose intravenous steroids, intravenous fluids, and eltroxine. He progressively regained consciousness within 72 h. Physical examination at this time showed a pale sallow complexion. He was blind in the right eye with temporal hemianopia in the left eye. Visual acuity of left eye was 6/60. Fundus examination showed primary optic atrophy in both eyes. Hormonal profile revealed hyperprolactinemia (4,000 IU/l). The patient also developed polyuria with a urinary volume of 9-12 l/day. He was treated with bromocriptine and prepared for surgery. A right frontal craniotomy using subfrontal approach was performed. Upon incising the diaphragma sellae 5-7 ml of necrotic material was excised. Postoperatively, the patient developed frank diabetes insipidus that needed aqueous vasopressin treatment. His visual acuity did not improve.
Haematoxylin and eosin-stained sections from the biopsy showed complete effacement of the pituitary structure by a dense lymphoplasmacytic infiltrate and foci of neutrophilic infiltration, necrosis, and fibrosis [Figure - 3]. No tumor could be identified. Stains for bacteria, fungi, Acid Fast Bacilli and spirochaetes were negative. Immunohistochemistry revealed a polyclonal lymphoid population with a mixture of T and B cells. A diagnosis of lymphocytic hypophysitis was considered. The patient was discharged on a maintenance dose of steroids and is currently on regular follow up.
Lymphocytic hypophysitis closely mimics a pituitary adenoma both clinically and radiologically. MR features suggestive of an inflammatory pituitary process include loss of hyperintense bright spot signal of the normal neurohypophysis, thickening of pituitary stalk and enlargement of neurohypophysis in cases where it is involved.[6] A review of literature revealed only two case reports of this association with lymphocytic hypophysitis (both in female patients),[3],[4] and one case with granulomatous hypophysitis (intrasellar tuberculoma).[7] Surgical intervention is required to establish the diagnosis and to reduce the size of the lesion to relieve the mass effect on adjacent structures. Prasad et al.[8] proposed a management paradigm for suspected cases of lymphocytic hypophysitis and advocated a trans sphenoidal stereotactic biopsy to achieve a tissue diagnosis, which might avoid the need for an open exploration of the sella.
The present case highlights the fact that apoplectic changes may complicate the course of inflammatory pituitary lesions, thus calling for a greater vigil and clinical judgment in management of these patients.
| » References | |  |
| 1. | Sautner D, Saeger W, Ludecke D K, Jansen V, Puchner MJA. Hypophysitis in surgical and autoptical specimens. Acta Neuropathol 1995;90:637-44.  |
| 2. | Goudie RB, Pickerton PH. Anterior hypophysitis and Hashimoto's disease in a young woman. J Path Bacteriol1962; 83:583-85. |
| 3. | Caturegli P, Newschaffer C, Olivi A, Pomper MG, Burger PC, Rose NR. Auto immune hypophysitis. Endocr Rev 2005 Jan 5; (E pub ahead of print) |
| 4. | Dan NG, Feiner RID, Houang MTW, Turner JJ. Pituitary apoplexy in association with lymphocytic hypophysitis. J Clin Neurosc 2002; 9:577-80. |
| 5. | Lee MS, Pless M. Apoplectic lymphocytic hypophysitis-case report. J Neurosurg; 2003; 98:183-85. |
| 6. | Imura H, Nakao K, Shimatsu A,Ogawa Y, Sando T, Fujisawa I, Yamabe H. Lymphocytic infundibuloneurohypophysitis as a cause of central diabetes insipidus. N Engl J Med 1993; 329:683-89. |
| 7. | Arunkumar MJ, Rajshekhar V. Intrasellar tuberculoma presenting as pituitary apoplexy. Neurol India 2001; 49:407-10. [PUBMED] [FULLTEXT] |
| 8. | Prasad A, Madan VS, Sethi PK, Prasad ML, Buxi TBS, Kanwar CK. Lymphocytic hypophysitis-can open exploration of the sella be avoided. Br J Neurosurg 1991; 5: 639-42. |
Figures
[Figure - 1], [Figure - 2], [Figure - 3]
| This article has been cited by | | 1 |
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From pituitary expansion to empty sella: Disease progression in a mouse model of autoimmune hypophysitis |
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| Lupi, I. and Zhang, J. and Gutenberg, A. and Landek-Salgado, M. and Tzou, S.-C. and Mori, S. and Caturegli, P. | | Endocrinology. 2011; 152(11): 4190-4198 | | [Pubmed] | | | 4 |
Diagnosis and treatment of autoimmune hypophysitis: A short review |
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| Lupi, I. and Manetti, L. and Raffaelli, V. and Lombardi, M. and Cosottini, M. and Iannelli, A. and Basolo, F. and Proietti, A. and Bogazzi, F. and Caturegli, P. and Martino, E. | | Journal of Endocrinological Investigation. 2011; 34(8 SUPPL.): e245-e252 | | [Pubmed] | | | 5 |
Gamma Knife surgery for lymphocytic hypophysitis |
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| Dibyendu Kumar Ray,Chun Po Yen,Mary Lee Vance,Edward R. Laws,Beatriz Lopes,Jason P. Sheehan | | Journal of Neurosurgery. 2010; 112(1): 118 | | [Pubmed] | [DOI] | | | 6 |
Gamma knife surgery for lymphocytic hypophysitis |
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| Ray, D.K., Yen, C.P., Vance, M.L., Laws, E.R., Lopes, B., Sheehan, J.P. | | Journal of Neurosurgery. 2010; 112(1): 118-121 | | [Pubmed] | |
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