Literature DB >> 31748928

Granulomatous hypophysitis causing compression of the internal carotid arteries reversible with azathioprine and rituximab treatment.

Pauline Gendreitzig1, Jürgen Honegger2, Marcus Quinkler3.   

Abstract

INTRODUCTION: Compression of the internal carotid artery (ICA) in the cavernous sinus area is a rare event and is mostly associated with pituitary adenomas and meningiomas. Other causes of ICA compression are less well known. We present a rare case of granulomatous hypophysitis causing compression of the ICA, which was treated successfully with immune-suppressive agents.
METHODS: The electronic database MEDLINE (PubMed) was searched systematically and other cases with ICA compression were identified and analyzed.
RESULTS: A female patient with a history of two previous transsphenoidal operations for suspected pituitary adenoma and post-operative complete pituitary insufficiency presented with severe headaches, nausea, fatigue, and diplopia. Pituitary MRI scan suggested relapse of the pituitary lesion with atypical bilateral infiltration of cavernous sinuses and compression of ICAs. After histological reevaluation of her previous pituitary operations, granulomatous hypophysitis was diagnosed. Treatment was started with high doses of prednisolone. With decreasing doses of prednisolone, symptoms recurred, and azathioprine was started, followed by administration of rituximab resulting in clinical recovery and regression of ICA compression. Literature analysis disclosed 36 case reports with ICA compression in the cavernous sinus region (12 pituitary adenoma, 6 meningioma, 7 hypophysitis, 5 other tumors, and 4 other etiologies). Two cases of hypophysitis recovered completely; five cases improved only partly.
CONCLUSION: In the case of ICA compression, clinical signs, onset of symptoms, radiological findings and pituitary insufficiencies should be thoroughly evaluated, and hypophysitis should be considered as a possible cause. In our patient, treatment with azathioprine and, finally, rituximab was successful.

Entities:  

Keywords:  Azathioprine; Meningioma; Pituitary adenoma; Prednisolone; Rituximab

Mesh:

Substances:

Year:  2020        PMID: 31748928     DOI: 10.1007/s11102-019-01005-2

Source DB:  PubMed          Journal:  Pituitary        ISSN: 1386-341X            Impact factor:   4.107


  40 in total

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Authors:  Seung-Ho Yang; Kwan-Sung Lee; Kyo-Young Lee; Sang Won Lee; Yong-Kil Hong
Journal:  J Korean Med Sci       Date:  2008-12-23       Impact factor: 2.153

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  2 in total

1.  Infiltrative lymphocytic hypophysitis successfully treated with rituximab and mycophenolate mofetil.

Authors:  Mike Lin; Venessa Tsang; Janice Brewer; Roderick Clifton-Bligh; Matti L Gild
Journal:  Endocrinol Diabetes Metab Case Rep       Date:  2020-07-29

2.  A Remarkable Response of Granulomatous Hypophysitis to Infliximab in a Patient With a Background of Crohn's Disease-A Case Report.

Authors:  Bahar K Force; Tiphanie P Vogel; Dang M Nguyen; Kent A Heck; Sherly Sebastian; Mas Takashima; Daniel Yoshor; Susan L Samson
Journal:  Front Endocrinol (Lausanne)       Date:  2020-05-29       Impact factor: 5.555

  2 in total

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