Sasan Darius Adib, Daniel Kopf, Brigitte Ruh-Daikeler, Rudi Beschorner, Antje Bornemann, Florian Hennersdorf, Jürgen Honegger
{"title":"Carotid artery constriction in autoimmune hypophysitis: three case reports and literature review.","authors":"Sasan Darius Adib, Daniel Kopf, Brigitte Ruh-Daikeler, Rudi Beschorner, Antje Bornemann, Florian Hennersdorf, Jürgen Honegger","doi":"10.1530/EC-25-0120","DOIUrl":null,"url":null,"abstract":"<p><strong>Objective: </strong>An intracavernous internal carotid artery constriction or occlusion (ICAc/o) has been considered an extremely rare finding in autoimmune hypophysitis (AiHy). This study aimed to analyse predictive factors for the occurrence of ICAc/o in AiHy.</p><p><strong>Design: </strong>Retrospective analysis of three of our own cases and 16 published cases.</p><p><strong>Methods: </strong>Among 15 surgically treated patients with AiHy, we identified three cases with ICAc/o via time-of-flight magnetic resonance angiography (TOF MRA) or computed tomography angiography (CTA). In addition, 16 published cases with AiHy and ICAc/o were identified via the literature search. Clinical features, treatment, and outcomes were evaluated.</p><p><strong>Results: </strong>TOF MRA revealed complete bilateral ICA occlusion (ICAo) in case 1 and incomplete bilateral ICA constriction (ICAc) in case 2. In the third case, left-sided ICAo was confirmed by CTA. None of our three patients with AiHy complicated by ICAc/o suffered brain infarction or neurological deficits. All three cases exhibited a parasellar T2 dark sign and strong dural enhancement. With our three cases included, seven of 19 published cases (36.8%) showed complete bilateral ICAo. Among these, four presented with ischaemic stroke. Eight of 19 patients (42.1%) presented with cranial nerve palsy. While all patients presented with hypopituitarism, only five had arginine vasopressin (AVP) deficiency. Interestingly, 11 patients had a recurrent course of hypophysitis.</p><p><strong>Conclusion: </strong>ICAc/o caused by AiHy appears to be more frequent than previously reported. Special attention should be paid to the carotid arteries in AiHy because of the potentially deleterious complication of ICAc/o. Cranial nerve palsy, a parasellar T2 dark sign, strong perisellar dural enhancement, and a recurrent course of hypophysitis can be considered warning signs of the occurrence of ICAc/o.</p>","PeriodicalId":11634,"journal":{"name":"Endocrine Connections","volume":" ","pages":""},"PeriodicalIF":2.8000,"publicationDate":"2025-05-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12120927/pdf/","citationCount":"0","resultStr":null,"platform":"Semanticscholar","paperid":null,"PeriodicalName":"Endocrine Connections","FirstCategoryId":"3","ListUrlMain":"https://doi.org/10.1530/EC-25-0120","RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"2025/6/1 0:00:00","PubModel":"Print","JCR":"Q3","JCRName":"ENDOCRINOLOGY & METABOLISM","Score":null,"Total":0}
引用次数: 0
Abstract
Objective: An intracavernous internal carotid artery constriction or occlusion (ICAc/o) has been considered an extremely rare finding in autoimmune hypophysitis (AiHy). This study aimed to analyse predictive factors for the occurrence of ICAc/o in AiHy.
Design: Retrospective analysis of three of our own cases and 16 published cases.
Methods: Among 15 surgically treated patients with AiHy, we identified three cases with ICAc/o via time-of-flight magnetic resonance angiography (TOF MRA) or computed tomography angiography (CTA). In addition, 16 published cases with AiHy and ICAc/o were identified via the literature search. Clinical features, treatment, and outcomes were evaluated.
Results: TOF MRA revealed complete bilateral ICA occlusion (ICAo) in case 1 and incomplete bilateral ICA constriction (ICAc) in case 2. In the third case, left-sided ICAo was confirmed by CTA. None of our three patients with AiHy complicated by ICAc/o suffered brain infarction or neurological deficits. All three cases exhibited a parasellar T2 dark sign and strong dural enhancement. With our three cases included, seven of 19 published cases (36.8%) showed complete bilateral ICAo. Among these, four presented with ischaemic stroke. Eight of 19 patients (42.1%) presented with cranial nerve palsy. While all patients presented with hypopituitarism, only five had arginine vasopressin (AVP) deficiency. Interestingly, 11 patients had a recurrent course of hypophysitis.
Conclusion: ICAc/o caused by AiHy appears to be more frequent than previously reported. Special attention should be paid to the carotid arteries in AiHy because of the potentially deleterious complication of ICAc/o. Cranial nerve palsy, a parasellar T2 dark sign, strong perisellar dural enhancement, and a recurrent course of hypophysitis can be considered warning signs of the occurrence of ICAc/o.
期刊介绍:
Endocrine Connections publishes original quality research and reviews in all areas of endocrinology, including papers that deal with non-classical tissues as source or targets of hormones and endocrine papers that have relevance to endocrine-related and intersecting disciplines and the wider biomedical community.