Carotid artery constriction in autoimmune hypophysitis: three case reports and literature review
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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 occurrence of ICAc/o in AiHy.
Design: Retrospective analysis of three 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 a literature search. Clinical features, treatment, and outcomes were evaluated.
Results: TOF MRA revealed a complete bilateral ICA occlusion (ICAo) in Case 1 and an 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 a strong dural enhancement.
With three own 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.
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