Postpartum hypothalamic adrenal insufficiency with remission: A rare case

作者：Akehi Yuko; Hashimoto Yoko; Meren Junko; Tanabe Makito; Nomiyama Takashi; Yanase Toshihiko^*

来源：Endocrine Journal, 2017, 64(2): 157-162.

DOI：10.1507/endocrj.ej16-0066

• Autoimmunity
• Hypothalamic adrenal insufficiency
• Remission
• Basedow's disease
• Sheehan's syndrome

A 37-year-old female patient was hospitalized because of general fatigue and loss of axillary and pubic hair after massive bleeding at delivery of her third child. The basal levels of both plasma adrenocorticotropin hormone (ACTH) and serum cortisol were very low, 5.2 pg/mL and 1.9 mu g/dL, respectively. Based on the fact that ACTH showed a low response to insulin tolerance test and a normal response to corticotropin-releasing hormone (CRH), she was diagnosed with hypothalamic adrenal insufficiency. No organic lesions were found in the hypothalamic-pituitary region by pituitary MRI and hydrocortisone therapy was instituted. Basedow's disease was also discovered and treated with methimazole, and thyroid function returned to normal. Surprisingly, adrenal insufficiency gradually resolved, making it possible to stop hydrocortisone therapy 2 years from the onset of disease. To our knowledge, there are no previous case reports discussing the remission of hypothalamic adrenal insufficiency. The etiology of the unusual clinical course of this case remains unclear and we discussed several possibilities of the pathogenesis.

• 出版日期2017