肉芽腫性多血管炎致中樞性尿崩癥一例并文獻復習_Chinese Journal of Respiratory and Critical Care Medicine

Authors：

宋慧慧 , 呂游 ,  唐昊

第二軍醫大學長征醫院呼吸與危重癥醫學科（上海 200003）;

Corresponding?author：

唐昊, Email: 13816033045@126.com

Keywords：

肉芽腫性多血管炎; 尿崩癥

DOI：

10.7507/1671-6205.201903059

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目的探討肉芽腫性多血管炎累及垂體的臨床表現、影像學特點、治療及預后。方法對 1 例確診的肉芽腫性多血管炎致中樞性尿崩癥患者的臨床資料結合文獻復習進行分析，總結其臨床特點、治療及預后。結果本例患者為女性，66 歲，以耳、鼻受損為首發癥狀，合并肺部、腎臟病變，垂體受累表現為中樞性尿崩癥及垂體后葉高信號消失，糖皮質激素誘導治療后尿崩癥緩解。結合文獻復習，肉芽腫性多血管炎是最常出現垂體病變的血管炎類型，發病率 1% 左右，好發于女性，多數表現為中樞性尿崩癥，其次是腺垂體功能減退、高泌乳素血癥。典型的垂體磁共振成像病變征象為 T1 加權相上垂體后葉高信號消失。既往多采用糖皮質激素聯合環磷酰胺的誘導緩解方案，但因治療不及時多數患者不能恢復正常垂體功能。結論肉芽腫性多血管炎累及垂體的情況極為罕見且起病隱匿。尿崩癥通常為首發癥狀或與耳鼻喉癥狀伴行，肺、腎臟受累癥狀輕、出現遲。早期診斷和及時治療有利于減少垂體的不可逆損傷，保存正常垂體功能。

Citation： 宋慧慧, 呂游, 唐昊. 肉芽腫性多血管炎致中樞性尿崩癥一例并文獻復習. 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.	李靜, 陳正賢, 郭紀全, 等. 韋格內肉芽腫 4 例診治分析. 實用醫學雜志, 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. 李靜, 陳正賢, 郭紀全, 等. 韋格內肉芽腫 4 例診治分析. 實用醫學雜志, 2005, 21(21): 2396-2397.

Chinese Journal of Respiratory and Critical Care Medicine

肉芽腫性多血管炎致中樞性尿崩癥一例并文獻復習

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