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以颅内多器官受累为表现的自身免疫性垂体炎:三例报告并文献复习

Autoimmune hypophysitis presenting with intracranial multi-organ involvement: three case reports and review of the literature.

作者信息

Kanoke Atsushi, Ogawa Yoshikazu, Watanabe Mika, Kumabe Toshihiro, Tominaga Teiji

机构信息

Department of Neurosurgery, Kohnan Hospital, 4-20-1 Nagamachiminami, Taihaku-ku, Sendai 982-8523, Miyagi, Japan.

出版信息

BMC Res Notes. 2013 Dec 28;6:560. doi: 10.1186/1756-0500-6-560.

DOI:10.1186/1756-0500-6-560
PMID:24373428
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC3877864/
Abstract

BACKGROUND

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. 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.

CONCLUSION

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. 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.

摘要

背景

自身免疫性垂体炎极少扩散至垂体组织以外的邻近器官,原因不明,仅有5例垂体炎扩散至海绵窦的报道。

病例介绍

3例患者出现非感染性垂体炎扩散至垂体组织以外并累及海绵窦。所有3例均经经蝶窦手术进行组织学确诊,术后采用脉冲剂量类固醇治疗后患者均有显著改善,包括磁共振成像显示双侧下丘脑异常信号强度消失、颈内动脉严重狭窄缓解以及肿胀的垂体组织恢复正常。3例中有2例符合免疫球蛋白G4相关疾病的组织学标准,尽管所有患者血清免疫球蛋白G4水平均不高。

结论

垂体炎这种不寻常地扩散至邻近器官的真正意义尚未完全明了,但在这种不寻常的扩散方式中,其发生机制可能因炎症发生时间而异。即使对于颈内动脉进行性严重狭窄且视力迅速恶化的患者,脉冲剂量类固醇治疗也取得了显著良好的效果。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/14a6/3877864/de8858493d00/1756-0500-6-560-6.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/14a6/3877864/416968e5287c/1756-0500-6-560-1.jpg
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https://cdn.ncbi.nlm.nih.gov/pmc/blobs/14a6/3877864/27cd37771470/1756-0500-6-560-3.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/14a6/3877864/9af3f19c923d/1756-0500-6-560-4.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/14a6/3877864/90fa6fdacdec/1756-0500-6-560-5.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/14a6/3877864/de8858493d00/1756-0500-6-560-6.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/14a6/3877864/416968e5287c/1756-0500-6-560-1.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/14a6/3877864/026f7078b9c1/1756-0500-6-560-2.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/14a6/3877864/27cd37771470/1756-0500-6-560-3.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/14a6/3877864/9af3f19c923d/1756-0500-6-560-4.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/14a6/3877864/90fa6fdacdec/1756-0500-6-560-5.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/14a6/3877864/de8858493d00/1756-0500-6-560-6.jpg

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