Abstract
BackgroundAutoimmune hypophysitis very rarely spreads to nearby organs outside the pituitary tissue, for unknown reasons, with only 5 reported cases of hypophysitis spreading over the cavernous sinus.Case presentationThree patients presented with cases of non-infectious hypophysitis spreading outside the pituitary tissue over the cavernous sinus. All three cases were diagnosed with histological confirmation by transsphenoidal surgery, and the patients showed remarkable improvement with postoperative pulse dose steroid therapy, including disappearance of abnormal signal intensities in the bilateral hypothalami on magnetic resonance imaging, resolution of severe stenosis of the internal carotid artery, and normalization of swollen pituitary tissues. Two of 3 cases fulfilled the histological criteria of immunoglobulin G4-related disease, although none of the patients had high serum immunoglobulin G4 level.ConclusionThe true implications of such unusual spreading of hypophysitis to nearby organs are not fully understood, but the mechanism of occurrence might vary according to the timing of inflammation in this unusual mode of spreading. Pulse dose steroid therapy achieved remarkably good outcomes even in the patient with progressive severe stenosis of the internal carotid artery and rapid visual deterioration.
Highlights
Autoimmune hypophysitis very rarely spreads to nearby organs outside the pituitary tissue, for unknown reasons, with only 5 reported cases of hypophysitis spreading over the cavernous sinus.Case presentation: Three patients presented with cases of non-infectious hypophysitis spreading outside the pituitary tissue over the cavernous sinus
The true implications of such unusual spreading of hypophysitis to nearby organs are not fully understood, but the mechanism of occurrence might vary according to the timing of inflammation in this unusual mode of spreading
Autoimmune hypophysitis is classified into subtypes based on the histological findings into granulomatous hypophysitis, lymphocytic hypophysitis, and xanthomatous hypophysitis, or based on the site of involvement or endocrinological behavior into adenohypophysitis, infundibulo-neurohypophysitis, and panhypophysitis [1]
Summary
The present three cases of autoimmune hypophysitis spread outside the pituitary tissue with severe fibrosis. Pulse dose steroid therapy achieved remarkably good outcomes even in the patient with progressive severe stenosis of the internal carotid artery and rapid visual deterioration. Consent Written informed consent was obtained from the three patients for publication of this case report and accompanying images. Authors’ contributions AK analyzed the patient data regarding the endocrinological outcome, and was a major contributor in writing the manuscript. YO performed transsphenoidal biopsy of all the patients. TK performed tumor removal in the first surgery of Case 3. All authors read and approved the final manuscript. Author details 1Department of Neurosurgery, Kohnan Hospital, 4-20-1 Nagamachiminami, Taihaku-ku, Sendai 982-8523, Miyagi, Japan. Author details 1Department of Neurosurgery, Kohnan Hospital, 4-20-1 Nagamachiminami, Taihaku-ku, Sendai 982-8523, Miyagi, Japan. 2Department of Pathology, Tohoku University Graduate School of Medicine, Sendai, Miyagi, Japan. 3Department of Neurosurgery, Tohoku University Graduate School of Medicine, Sendai, Miyagi, Japan
Sign-in/Register to view highlights & summary Sign-in/Register to access full text options 
Talk to us
Join us for a 30 min session where you can share your feedback and ask us any queries you have
Schedule a callDisclaimer: All third-party content on this website/platform is and will remain the property of their respective owners and is provided on "as is" basis without any warranties, express or implied. Use of third-party content does not indicate any affiliation, sponsorship with or endorsement by them. Any references to third-party content is to identify the corresponding services and shall be considered fair use under The CopyrightLaw.
