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以严重低钠血症和横纹肌溶解症为表现的孤立性促肾上腺皮质激素缺乏症:病例报告及文献综述

Isolated Adrenocorticotropic Hormone Deficiency Presenting with Severe Hyponatremia and Rhabdomyolysis: A Case Report and Literature Review.

作者信息

Komatsu Takeshi, Ohara Nobumasa, Hirota Naoko, Yoneoka Yuichiro, Tani Takashi, Terajima Keishi, Ozawa Tetsutaro, Sone Hirohito

机构信息

Department of Endocrinology and Metabolism, Uonuma Institute of Community Medicine, Niigata University Medical and Dental Hospital, Niigata City, Niigata, Japan.

Department of Internal Medicine, Niigata Prefectural Tokamachi Hospital, Niigata City, Niigata, Japan.

出版信息

Am J Case Rep. 2019 Dec 12;20:1857-1863. doi: 10.12659/AJCR.918427.

Abstract

BACKGROUND Isolated adrenocorticotropic hormone deficiency (IAD) is a rare disorder characterized by central adrenal insufficiency (AI) but normal secretion of pituitary hormones other than adrenocorticotropic hormone. IAD usually presents with unspecific symptoms of AI, such as anorexia and fatigue, but some patients present with a variety of atypical manifestations. Rhabdomyolysis is a potentially life-threatening clinical syndrome caused by skeletal muscle injury with the release of muscle cell contents into the circulation. A wide variety of disorders can cause rhabdomyolysis. Herein, we report an unusual case of IAD presenting with hyponatremia and rhabdomyolysis. CASE REPORT A 67-year-old Japanese woman with a 2-month history of anorexia and fatigue was diagnosed with severe hyponatremia (serum sodium, 118 mEq/L) and rhabdomyolysis (serum creatine phosphokinase, 6968 IU/L), after 2 days of vomiting and muscle weakness. Physical and laboratory findings did not show dehydration or peripheral edema. Her rhabdomyolysis resolved with normalization of serum sodium levels during administration of sodium chloride. However, her anorexia and fatigue remained unresolved. After reducing the amount of sodium chloride administered, the patient still had hyponatremia. Detailed endocrinological examinations indicated IAD; her hyponatremia was associated with inappropriately high plasma arginine vasopressin levels. The patient received corticosteroid replacement therapy, which resolved her anorexia, fatigue, excessive arginine vasopressin, and hyponatremia. CONCLUSIONS This case highlights the importance of considering the possibility of central AI in patients with hyponatremia and excessive arginine vasopressin levels. In addition, rhabdomyolysis associated with hyponatremia can be an important manifestation of IAD.

摘要

背景

孤立性促肾上腺皮质激素缺乏症(IAD)是一种罕见的疾病,其特征为中枢性肾上腺功能不全(AI),但除促肾上腺皮质激素外的垂体激素分泌正常。IAD通常表现为AI的非特异性症状,如厌食和疲劳,但一些患者会出现各种非典型表现。横纹肌溶解是一种由骨骼肌损伤导致肌肉细胞内容物释放到循环系统中引起的潜在危及生命的临床综合征。多种疾病可导致横纹肌溶解。在此,我们报告一例表现为低钠血症和横纹肌溶解的不寻常IAD病例。病例报告:一名67岁的日本女性,有2个月的厌食和疲劳病史,在出现呕吐和肌肉无力2天后,被诊断为严重低钠血症(血清钠,118 mEq/L)和横纹肌溶解(血清肌酸磷酸激酶,6968 IU/L)。体格检查和实验室检查未显示脱水或外周水肿。在给予氯化钠治疗期间,随着血清钠水平恢复正常,她的横纹肌溶解得到缓解。然而,她的厌食和疲劳仍未缓解。减少氯化钠的给药量后,患者仍有低钠血症。详细的内分泌检查表明为IAD;她的低钠血症与血浆精氨酸加压素水平异常升高有关。患者接受了皮质类固醇替代治疗,这使她的厌食、疲劳、精氨酸加压素过多和低钠血症得到缓解。结论:该病例强调了在低钠血症和精氨酸加压素水平过高的患者中考虑中枢性AI可能性的重要性。此外,与低钠血症相关的横纹肌溶解可能是IAD的一个重要表现。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/0b65/6931390/0c4841a4c888/amjcaserep-20-1857-g001.jpg

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