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山东大学学报 (医学版) ›› 2022, Vol. 60 ›› Issue (8): 98-102.doi: 10.6040/j.issn.1671-7554.0.2022.0699

• 临床医学 • 上一篇    下一篇

以骨痛为首发表现的原发性胆汁性胆管炎1例

陈诗鸿,姜冬青,庄向华,李晓博,潘喆,孙爱丽,娄能俊,王殿辉,杜娇娇,宋玉文   

  1. 山东大学第二医院内分泌科, 山东 济南 250033
  • 发布日期:2022-07-27
  • 通讯作者: 宋玉文. E-mail:songyuwen01@126.com
  • 基金资助:
    山东大学齐鲁医学院本科教学改革与研究项目(qlyxjy-201882)

Primary biliary cholangitis with bone pain as the main manifestation: a case report

CHEN Shihong, JIANG Dongqing, ZHUANG Xianghua, LI Xiaobo, PAN Zhe, SUN Aili, LOU Nengjun, WANG Dianhui, DU Jiaojiao, SONG Yuwen   

  1. Department of Endocrinology, The Second Hospital of Shandong University, Jinan 250033, Shandong, China
  • Published:2022-07-27

摘要: 目的 原发性胆汁性胆管炎(PBC)是一种少见的自身免疫性肝病,也是继发性骨质疏松症的罕见病因。本文通过报告1例以骨痛起病的PBC病例,旨在提高临床医师对该病的重视。 方法 纳入1例以骨痛为主要表现的老年女性,评估患者实验室检查指标、骨密度、骨骼X线影像特点,并通过复习文献,总结PBC合并骨质疏松症的临床特点。 结果 患者为61岁女性,双髋、双膝疼痛1年余,双能X线骨密度检查提示骨质疏松症。患者无乏力、皮肤瘙痒、黄染等胆道梗阻表现,肝功示胆管酶γ-谷氨酰转肽酶(GGT)、碱性磷酸酶(ALP)升高, 抗线粒体抗体Ⅱ型(AMA-M2)阳性,提示患者为PBC导致的继发性骨质疏松症。 结论 PBC临床表现隐匿,以骨痛、骨质疏松症起病少见。骨质疏松症患者如合并胆管酶升高、AMA-M2阳性,应考虑该病可能。

关键词: 原发性胆汁性胆管炎, 继发性骨质疏松症, 骨痛, 双膦酸盐

Abstract: Objective Primary biliary cholangitis(PBC)is a rare autoimmune liver disease, which is one of the rare causes of secondary osteoporosis. In this paper, we will report a case of PBC with bone pain as the main manifestation, in order to arouse clinicians attention to this disease. Methods A case of elderly women with bone pain as the main manifestation was enrolled, and the results of laboratory examinations, bone mineral density and X-ray image of bones were evaluated. The clinical characteristics of PBC with osteoporosis were summarized by reviewing the literature. Results The patient was a 61-year-old female with pain in both hips and knees for more than 1 year. Dual-energy X-ray absorptiometry indicated osteoporosis. The patient showed no symptoms of biliary tract obstruction such as fatigue, skin itching, or yellow staining. However, laboratory examinations showed elevated biliary tract enzyme, including gamma-glutamyl transferase(GGT)and alkaline phosphatase(ALP). Her anti-mitochondrial antibody type Ⅱ(AMA-M2)was positive. These results indicated that her osteoporosis was probably caused by PBC. Conclusion The clinical manifestations of PBC are insidious. PBC should be considered in patients with osteoporosis, especially when the bile duct enzymes are elevated and AMA-M2 is positive.

Key words: Primary biliary cholangitis, Secondary osteoporosis, Bone pain, Bisphosphonates

中图分类号: 

  • R575.7
[1] Carey EJ, Ali AH, Lindor KD. Primary biliary cirrhosis [J]. Lancet, 2015, 386(10003): 1565-1575.
[2] Lleo A, Leung PSC, Hirschfield GM, et al. The pathogenesis of primary biliary cholangitis: a comprehensive review [J]. Semin Liver Dis, 2020, 40(1): 34-48.
[3] 陈成伟, 成军, 窦晓光, 等. 原发性胆汁性肝硬化(又名原发性胆汁性胆管炎)诊断和治疗共识(2015)[J]. 临床肝胆病杂志, 2015, 31(12):1980-1988.
[4] Angulo P. Strengthening the bones in primary biliary cirrhosis [J]. Hepatology, 2013, 58(6): 1871-1873.
[5] 夏维波, 章振林, 林华, 等. 原发性骨质疏松症诊疗指南(2017)[J]. 中华骨质疏松和骨矿盐疾病杂志, 2017, 10(5): 413-444.
[6] Liu H, Liu Y, Wang L, et al. Prevalence of primary biliary cirrhosis in adults referring hospital for annual health check-up in Southern China [J]. BMC Gastroenterol, 2010, 10: 100. doi: 10.1186/1471-230X-10-100.
[7] 张奉春, 王立, 帅宗文, 等. 原发性胆汁性胆管炎诊疗规范(2021)[J]. 中华内科杂志, 2021 60(8): 709-715.
[8] Glass LM, Su GL. Metabolic bone disease in primary biliary cirrhosis [J]. Gastroenterol Clin North Am, 2016, 45(2): 333-343.
[9] Raszeja-Wyszomirska J, Miazgowski T. Osteoporosis in primary biliary cirrhosis of the liver [J]. Prz Gastroenterol, 2014, 9(2): 82-87.
[10] Chalifoux SL, Konyn PG, Choi G, et al. Extrahepatic manifestations of primary biliary cholangitis [J]. Gut Liver, 2017, 11(6): 771-780.
[11] Liao CY, Chung CH, Chu P, et al. Increased risk of osteoporosis in patients with primary biliary cirrhosis [J]. PLoS One, 2018, 13(3): e0194418. doi: 10.1371/journal.pone.0194418.
[12] Guañabens N, Cerdá D, Monegal A, et al. Low bone mass and severity of cholestasis affect fracture risk in patients with primary biliary cirrhosis [J]. Gastroenterology, 2010, 138(7): 2348-2356.
[13] Lin CY, Cheng YT, Chang ML, et al. The extrahepatic events of Asian patients with primary biliary cholangitis: a 30-year cohort study [J]. Sci Rep, 2019, 9(1): 7577. doi: 10.1038/s41598-019-44081-x.
[14] Schmidt T, Schmidt C, Schmidt FN, et al. Disease duration and stage influence bone microstructure in patients with primary biliary cholangitis [J]. J Bone Miner Res, 2018, 33(6): 1011-1019.
[15] Tang R, Wei Y, Li Z, et al. A common variant in CLDN14 is associated with primary biliary cirrhosis and bone mineral density [J]. Sci Rep, 2016, 6:19877. doi: 10.1038/srep19877.
[16] Seki A, Ikeda F, Miyatake H, et al. Risk of secondary osteoporosis due to lobular cholestasis in non-cirrhotic primary biliary cholangitis [J]. J Gastroenterol Hepatol, 2017, 32(9): 1611-1616.
[17] Saeki C, Oikawa T, Kanai T, et al. Relationship between osteoporosis, sarcopenia, vertebral fracture, and osteosarcopenia in patients with primary biliary cholangitis [J]. Eur J Gastroenterol Hepatol, 2021, 33(5): 731-737.
[18] Jeong HM, Kim DJ. Bone diseases in patients with chronic liver disease [J]. Int J Mol Sci, 2019, 20(17): 4270. doi: 10.3390/ijms20174270.
[19] Ruiz-Gaspà S, Guañabens N, Jurado S, et al. Bilirubin and bile acids in osteocytes and bone tissue. potential role in the cholestatic-induced osteoporosis [J]. Liver Int, 2020, 40(11): 2767-2775.
[20] Silvia RG, Nuria G, Susana J, et al. Bile acids and bilirubin effects on osteoblastic gene profile. Implications in the pathogenesis of osteoporosis in liver diseases [J]. Gene, 2020, 725: 144167. doi: 10.1016/j.gene.2019.144167.
[21] Schmidt T, Schwinge D, Rolvien T, et al. Th17 cell frequency is associated with low bone mass in primary sclerosing cholangitis [J]. J Hepatol, 2019, 70(5): 941-953.
[22] Gulamhusein AF, Hirschfield GM. Primary biliary cholangitis: pathogenesis and therapeutic opportunities [J]. Nat Rev Gastroenterol Hepatol. 2020, 17(2): 93-110.
[23] Danford CJ, Trivedi HD, Papamichael K, et al. Osteoporosis in primary biliary cholangitis [J]. World J Gastroenterol, 2018, 24(31): 3513-3520.
[24] Trivedi HD, Danford CJ, Goyes D, et al. Osteoporosis in primary biliary cholangitis: prevalence, impact and management challenges [J]. Clin Exp Gastroenterol, 2020, 13:17-24. doi: 10.2147/CEG.S204638.
[25] Lindor KD, Bowlus CL, Boyer J, et al. Primary biliary cholangitis: 2018 practice guidance from the American Association for the study of liver diseases [J]. Hepatology, 2019, 69(1): 394-419.
[26] European Association for the Study of the Liver. EASL Clinical Practice Guidelines: The diagnosis and management of patients with primary biliary cholangitis [J]. J Hepatol, 2017, 67(1): 145-172.
[27] Rudic JS, Giljaca V, Krstic MN, et al. Bisphosphonates for osteoporosis in primary biliary cirrhosis [J]. Cochrane Database Syst Rev, 2011,(12): CD009144. doi: 10.1002/14651858.CD009144.pub2.
[28] Guañabens N, Monegal A, Cerdá D, et al. Randomized trial comparing monthly ibandronate and weekly alendronate for osteoporosis in patients with primary biliary cirrhosis [J]. Hepatology, 2013, 58(6): 2070-2078.
[29] Rudic JS, Poropat G, Krstic MN, et al. Hormone replacement for osteoporosis in women with primary biliary cirrhosis [J]. Cochrane Database Syst Rev, 2011,(12): CD009146. doi: 10.1002/14651858.CD009146.pub2.
[30] Danford CJ, Ezaz G, Trivedi HD, et al. The pharmacologic management of osteoporosis in primary biliary cholangitis: a systematic review and meta-analysis [J]. J Clin Densitom, 2020, 23(2): 223-236.
[31] Xu X, Wang R, Wu R, et al. Trehalose reduces bone loss in experimental biliary cirrhosis rats via ERK phosphorylation regulation by enhancing autophagosome formation [J]. FASEB J, 2020, 34(6): 8402-8415.
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