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本文引用的文献

1
Systemic effects of hypothermia due to hypothalamic dysfunction after resection of a craniopharyngioma: case report and review of literature.颅咽管瘤切除术后下丘脑功能障碍所致低温的全身影响:病例报告及文献复习
Neuropediatrics. 2013 Jun;44(3):159-62. doi: 10.1055/s-0032-1327773. Epub 2012 Oct 9.
2
Involvement of hypothalamus autoimmunity in patients with autoimmune hypopituitarism: role of antibodies to hypothalamic cells.自身免疫性垂体功能减退症患者的下丘脑自身免疫:抗下丘脑细胞抗体的作用。
J Clin Endocrinol Metab. 2012 Oct;97(10):3684-90. doi: 10.1210/jc.2012-2269. Epub 2012 Aug 1.
3
Diabetes insipidus, panhypopituitarism, and severe mental status deterioration in a patient with chordoid glioma: case report and literature review.脊索样胶质瘤患者出现尿崩症、全垂体功能减退和严重精神状态恶化:病例报告及文献综述
Endocr Pract. 2009 Apr;15(3):240-5. doi: 10.4158/EP.15.3.240.
4
The spectrum and significance of primary hypophysitis.原发性垂体炎的谱系及意义
J Clin Endocrinol Metab. 2001 Mar;86(3):1048-53. doi: 10.1210/jcem.86.3.7265.
5
Role of the preoptic-anterior hypothalamus in thermoregulation and fever.视前区-下丘脑前部在体温调节和发热中的作用。
Clin Infect Dis. 2000 Oct;31 Suppl 5:S157-61. doi: 10.1086/317521.
6
Lymphocytic hypophysitis presenting with diabetes insipidus: case report and literature review.以尿崩症为表现的淋巴细胞性垂体炎:病例报告及文献复习
Endocr J. 1994 Feb;41(1):93-7. doi: 10.1507/endocrj.41.93.
7
Disturbances of the hypothalamic thermoregulation.下丘脑体温调节障碍。
Acta Neurochir (Wien). 1985;75(1-4):106-12. doi: 10.1007/BF01406330.

一例罕见的以体温调节异常为表现的自身免疫性垂体炎。

A rare case of autoimmune hypophysitis presenting as temperature dysregulation.

作者信息

Jain Ankur, Dhanwal Dinesh K

机构信息

Post Graduate Student, Department of Medicine, Maulana Azad Medical College , Bahadur Shah Zafar Marg, Delhi, India .

Director Professor, Department of Medicine, Maulana Azad Medical College , Bahadur Shah Zafar Marg, Delhi, India .

出版信息

J Clin Diagn Res. 2015 Feb;9(2):OD09-10. doi: 10.7860/JCDR/2015/11618.5574. Epub 2015 Feb 1.

DOI:10.7860/JCDR/2015/11618.5574
PMID:25859485
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC4378767/
Abstract

Autoimmune hypophysitis is a rare cause of pan-hypopituitarism. Hypothalamic involvement in autoimmune hypophysitis is rare and usually manifests as central diabetes insipidus due to antibodies against arginine vasopressin. Temperature dysregulation is occasionally seen with suprasellar tumours but has never been reported with hypophysitis. We report a case of a middle aged man who presented to us with the complaints of documented body temperature fluctuations since two months followed gradually by hoarseness of voice, sexual dysfunction and syncope. Examination was remarkable for postural hypotension, dry coarse skin and delayed ankle reflexes. Patient's hormone profile revealed pan-hypopituitarism and elevated titre of anti-TPO antibodies. Patient's work up for secondary causes of hypopituitarism was negative. MRI brain revealed typical findings of hypophysitis. Patient was suspected as a case of autoimmune hypophysitis and was immediately treated with prednisolone along with hormone replacement. Rapid response within 30 days was observed in the form of subsidence of temperature fluctuations, improvement in general well being, sexual function and repeat MRI done after one month which revealed a partial empty sella. Autoimmune hypophysitis as a potentially treatable cause of temperature dysregulation has been highlighted in this case.

摘要

自身免疫性垂体炎是全垂体功能减退的罕见病因。下丘脑受累于自身免疫性垂体炎较为罕见,通常表现为由于抗精氨酸加压素抗体导致的中枢性尿崩症。体温调节异常偶尔可见于鞍上肿瘤,但垂体炎从未有过相关报道。我们报告一例中年男性,自两个月前出现有记录的体温波动,随后逐渐出现声音嘶哑、性功能障碍和晕厥。检查发现明显的体位性低血压、皮肤干燥粗糙和踝反射延迟。患者的激素检查显示全垂体功能减退且抗甲状腺过氧化物酶抗体滴度升高。患者垂体功能减退继发原因的检查结果为阴性。脑部磁共振成像显示垂体炎的典型表现。患者被怀疑为自身免疫性垂体炎,并立即接受泼尼松龙治疗及激素替代治疗。在30天内观察到快速反应,表现为体温波动消退、总体健康状况改善、性功能改善,且一个月后复查磁共振成像显示部分空蝶鞍。本病例突出了自身免疫性垂体炎作为体温调节异常潜在可治疗病因的情况。