Granulomatous hypophysitis causing compression of the internal carotid arteries reversible with azathioprine and rituximab treatment
- 42 Downloads
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.
The electronic database MEDLINE (PubMed) was searched systematically and other cases with ICA compression were identified and analyzed.
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.
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.
KeywordsPrednisolone Azathioprine Rituximab Pituitary adenoma Meningioma
Compliance with ethical standards
Conflict of interest
The authors declare that there is no conflict of interest that could be perceived as prejudicing the impartiality of the research reported.
All procedures performed in studies involving human participants were in accordance with the ethical standards of the institutional and/or national research committee (Berliner Ärztekammer) and with the 1964 Helsinki declaration and its later amendments or comparable ethical standards. This article does not contain any studies with animals performed by any of the authors.
Informed consent was obtained from all individual participants included in the study.
- 14.Alzhrani G, Derrico N, Abou-Al-Shaar H, Couldwell WT (2019) Management of cavernous sinus meningioma presenting with cerebrovascular insufficiency secondary to cavernous carotid artery occlusion: report of 2 cases. Op. Neurosurg. Hagerstown, Md.) 16:503–513Google Scholar
- 26.Boia ER et al (2013) Non-keratinizing undifferentiated carcinoma of the nasopharynx. Roman J Morphol Embryol = Revue roumaine de morphologie et embryologie 54:839–843Google Scholar
- 28.Kojima T, Waga S (1982) Stenosis of the intracranial internal carotid artery by a craniopharyngioma: report of a case: No shinkei geka. Neurol Surg 10:777–782Google Scholar
- 39.Katsiveli P et al (2016) A complicated case of primary hypophysitis with bilateral intracavernous carotid artery occlusion. Hormones (Athens, Greece) 15:291–296Google Scholar