肉芽肿性多血管炎致中枢性尿崩症一例并文献复习_Chinese Journal of Respiratory and Critical Care Medicine

Authors：

宋慧慧 , 吕游 ,  唐昊

第二军医大学长征医院呼吸与危重症医学科（上海 200003）;

Corresponding author：

唐昊, Email: 13816033045@126.com

Keywords：

肉芽肿性多血管炎; 尿崩症

DOI：

10.7507/1671-6205.201903059

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目的探讨肉芽肿性多血管炎累及垂体的临床表现、影像学特点、治疗及预后。方法对 1 例确诊的肉芽肿性多血管炎致中枢性尿崩症患者的临床资料结合文献复习进行分析，总结其临床特点、治疗及预后。结果本例患者为女性，66 岁，以耳、鼻受损为首发症状，合并肺部、肾脏病变，垂体受累表现为中枢性尿崩症及垂体后叶高信号消失，糖皮质激素诱导治疗后尿崩症缓解。结合文献复习，肉芽肿性多血管炎是最常出现垂体病变的血管炎类型，发病率 1% 左右，好发于女性，多数表现为中枢性尿崩症，其次是腺垂体功能减退、高泌乳素血症。典型的垂体磁共振成像病变征象为 T1 加权相上垂体后叶高信号消失。既往多采用糖皮质激素联合环磷酰胺的诱导缓解方案，但因治疗不及时多数患者不能恢复正常垂体功能。结论肉芽肿性多血管炎累及垂体的情况极为罕见且起病隐匿。尿崩症通常为首发症状或与耳鼻喉症状伴行，肺、肾脏受累症状轻、出现迟。早期诊断和及时治疗有利于减少垂体的不可逆损伤，保存正常垂体功能。

Citation： 宋慧慧, 吕游, 唐昊. 肉芽肿性多血管炎致中枢性尿崩症一例并文献复习. Chinese Journal of Respiratory and Critical Care Medicine, 2020, 19(1): 70-74. doi: 10.7507/1671-6205.201903059 Copy

1.	Kapoor E, Cartin-Ceba R, Specks U, et al. Pituitary dysfunction in granulomatosis with polyangiitis: the Mayo Clinic experience. J Clin Endocrinol Metab, 2014, 99(11): 3988-3994.
2.	De Parisot A, Puechal X, Langrand C, et al. Pituitary involvement in granulomatosis with polyangiitis: report of 9 patients and review of the literature. Medicine (Baltimore), 2015, 94(16): e748.
3.	Peters JE, Gupta V, Saeed IT, et al. Severe localised granulomatosis with polyangiitis (Wegener's granulomatosis) manifesting with extensive cranial nerve palsies and cranial diabetes insipidus: a case report and literature review. BMC Neurol, 2018, 18(1): 59.
4.	Bando H, Iguchi G, Fukuoka H, et al. A diagnostic pitfall in IgG4-related hypophysitis: infiltration of IgG4-positive cells in the pituitary of granulomatosis with polyangiitis. Pituitary, 2015, 18(5): 722-730.
5.	Yong TY, Li JY, Amato L, et al. Pituitary involvement in Wegener's granulomatosis. Pituitary, 2008, 11(1): 77-84.
6.	Nishino H, Rubino FA, DeRemee RA, et al. Neurological involvement in Wegener's granulomatosis: an analysis of 324 consecutive patients at the Mayo Clinic. Ann Neurol, 1993, 33(1): 4-9.
7.	Yates M, Watts RA. EULAR/ERA-EDTA recommendations for the management of ANCA-associated vasculitis. Ann Rheum Dis, 2016, 75(9): 1583-1594.
8.	Ohashi K, Morishita M, Watanabe H, et al. Central diabetes insipidus in refractory antineutrophil cytoplasmic antibody-associated vasculitis. Intern Med, 2017, 56(21): 2943-2948.
9.	Tsuji H, Yoshifuji H, Fujii T, et al. Visceral disseminated varicella zoster virus infection after rituximab treatment for granulomatosis with polyangiitis. Mod Rheumatol, 2017, 27(1): 155-161.
10.	Baird SM, Pratap U, McLean C, et al. Rare presentation of Wegener's granulomatosis in the pituitary gland: Case report and literature review. Int J Surg Case Rep, 2017, 33: 24-26.
11.	Dutta P, Hayatbhat M, Bhansali A, et al. Wegener's granulomatosis presenting as diabetes insipidus. Exp Clin Endocrinol Diabetes, 2006, 114(9): 533-536.
12.	Duzgun N, Morris Y, Gullu S, et al. Diabetes insipidus presentation before renal and pulmonary features in a patient with Wegener's granulomatosis. Rheumatol Int, 2005, 26(1): 80-82.
13.	Garovic VD, Clarke BL, Chilson TS, et al. Diabetes insipidus and anterior pituitary insufficiency as presenting features of Wegener's granulomatosis. Am J Kidney Dis, 2001, 37(1): E5.
14.	Hurst NP, Dunn NA, Chalmers TM. Wegener's granulomatosis complicated by diabetes insipidus. Ann Rheum Dis, 1983, 42(5): 600-601.
15.	Katzman GL, Langford CA, Sneller MC, et al. Pituitary involvement by Wegener's granulomatosis: a report of two cases. Am J Neuroradiol, 1999, 20(3): 519-523.
16.	Muir BM, Hulett RL, Zorn JG. Wegener's granulomatosis complicated by central diabetes insipidus in a pediatric patient. Am J Roentgenol, 2004, 182(6): 1560-1562.
17.	Sampei S, Watanabe R, Ishii T, et al. Granulomatosis with polyangiitis preceded by central diabetes insipidus. Intern Med, 2014, 53(15): 1725-1726.
18.	Xue J, Wang H, Wu H, et al. Wegener's granulomatosis complicated by central diabetes insipidus and peripheral neutrophy with normal pituitary in a patient. Rheumatol Int, 2009, 29(10): 1213-1217.
19.	Czarnecki EJ, Spickler EM. MR demonstration of Wegener granulomatosis of the infundibulum, a cause of diabetes insipidus. AJNR Am J Neuroradiol, 1995, 16(4 Suppl): 968-970.
20.	Santoro SG, Guida AH, Furioso AE, et al. Panhypopituitarism due to Wegener's granulomatosis. Arq Bras Endocrinol Metabol, 2011, 55(7): 481-485.
21.	李静, 陈正贤, 郭纪全, 等. 韦格内肉芽肿 4 例诊治分析. 实用医学杂志, 2005, 21(21): 2396-2397.

1. Kapoor E, Cartin-Ceba R, Specks U, et al. Pituitary dysfunction in granulomatosis with polyangiitis: the Mayo Clinic experience. J Clin Endocrinol Metab, 2014, 99(11): 3988-3994.
2. De Parisot A, Puechal X, Langrand C, et al. Pituitary involvement in granulomatosis with polyangiitis: report of 9 patients and review of the literature. Medicine (Baltimore), 2015, 94(16): e748.
3. Peters JE, Gupta V, Saeed IT, et al. Severe localised granulomatosis with polyangiitis (Wegener's granulomatosis) manifesting with extensive cranial nerve palsies and cranial diabetes insipidus: a case report and literature review. BMC Neurol, 2018, 18(1): 59.
4. Bando H, Iguchi G, Fukuoka H, et al. A diagnostic pitfall in IgG4-related hypophysitis: infiltration of IgG4-positive cells in the pituitary of granulomatosis with polyangiitis. Pituitary, 2015, 18(5): 722-730.
5. Yong TY, Li JY, Amato L, et al. Pituitary involvement in Wegener's granulomatosis. Pituitary, 2008, 11(1): 77-84.
6. Nishino H, Rubino FA, DeRemee RA, et al. Neurological involvement in Wegener's granulomatosis: an analysis of 324 consecutive patients at the Mayo Clinic. Ann Neurol, 1993, 33(1): 4-9.
7. Yates M, Watts RA. EULAR/ERA-EDTA recommendations for the management of ANCA-associated vasculitis. Ann Rheum Dis, 2016, 75(9): 1583-1594.
8. Ohashi K, Morishita M, Watanabe H, et al. Central diabetes insipidus in refractory antineutrophil cytoplasmic antibody-associated vasculitis. Intern Med, 2017, 56(21): 2943-2948.
9. Tsuji H, Yoshifuji H, Fujii T, et al. Visceral disseminated varicella zoster virus infection after rituximab treatment for granulomatosis with polyangiitis. Mod Rheumatol, 2017, 27(1): 155-161.
10. Baird SM, Pratap U, McLean C, et al. Rare presentation of Wegener's granulomatosis in the pituitary gland: Case report and literature review. Int J Surg Case Rep, 2017, 33: 24-26.
11. Dutta P, Hayatbhat M, Bhansali A, et al. Wegener's granulomatosis presenting as diabetes insipidus. Exp Clin Endocrinol Diabetes, 2006, 114(9): 533-536.
12. Duzgun N, Morris Y, Gullu S, et al. Diabetes insipidus presentation before renal and pulmonary features in a patient with Wegener's granulomatosis. Rheumatol Int, 2005, 26(1): 80-82.
13. Garovic VD, Clarke BL, Chilson TS, et al. Diabetes insipidus and anterior pituitary insufficiency as presenting features of Wegener's granulomatosis. Am J Kidney Dis, 2001, 37(1): E5.
14. Hurst NP, Dunn NA, Chalmers TM. Wegener's granulomatosis complicated by diabetes insipidus. Ann Rheum Dis, 1983, 42(5): 600-601.
15. Katzman GL, Langford CA, Sneller MC, et al. Pituitary involvement by Wegener's granulomatosis: a report of two cases. Am J Neuroradiol, 1999, 20(3): 519-523.
16. Muir BM, Hulett RL, Zorn JG. Wegener's granulomatosis complicated by central diabetes insipidus in a pediatric patient. Am J Roentgenol, 2004, 182(6): 1560-1562.
17. Sampei S, Watanabe R, Ishii T, et al. Granulomatosis with polyangiitis preceded by central diabetes insipidus. Intern Med, 2014, 53(15): 1725-1726.
18. Xue J, Wang H, Wu H, et al. Wegener's granulomatosis complicated by central diabetes insipidus and peripheral neutrophy with normal pituitary in a patient. Rheumatol Int, 2009, 29(10): 1213-1217.
19. Czarnecki EJ, Spickler EM. MR demonstration of Wegener granulomatosis of the infundibulum, a cause of diabetes insipidus. AJNR Am J Neuroradiol, 1995, 16(4 Suppl): 968-970.
20. Santoro SG, Guida AH, Furioso AE, et al. Panhypopituitarism due to Wegener's granulomatosis. Arq Bras Endocrinol Metabol, 2011, 55(7): 481-485.
21. 李静, 陈正贤, 郭纪全, 等. 韦格内肉芽肿 4 例诊治分析. 实用医学杂志, 2005, 21(21): 2396-2397.

Chinese Journal of Respiratory and Critical Care Medicine

肉芽肿性多血管炎致中枢性尿崩症一例并文献复习

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