A Case of Adrenal Insufficiency Due to Acquired Hypothalamic CRH Deficiency.

Nishihara, Eijun; Kimura, Hironori; Ishimaru, T.; Kiriyama, Takeshi; Yokoyama, Naokata; Yamashita, Shunichi; Nagataki, Shigenobu

doi:10.1507/endocrj.44.121

Cited by 13 publications

(5 citation statements)

References 9 publications

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“…It has been reported that the autoimmune process may underlie the onset of hypothalamic AI [23][24][25], but autoimmune involvement was not observed in this study population. However, we did not investigate antihypothalamic and anti-pituitary antibodies in the present study.…”

Section: Discussioncontrasting

confidence: 68%

Clinical characteristics of patients with unexplainable hypothalamic disorder diagnosed by the corticotropin-releasing hormone challenge test: a retrospective study

et al. 2022

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Background The corticotropin-releasing hormone (CRH) challenge test can distinguish the disorders of the hypothalamus from those of the pituitary. However, the pathophysiology of hypothalamic disorder (HD) has not been fully understood. This study aimed to elucidate the clinical characteristics of patients with unexplainable HD, diagnosed by the CRH challenge test. Methods We retrospectively reviewed patients who underwent the CRH challenge test. Patients were categorized into four groups as follows: patients with peak serum cortisol ≥18 μg/dL were assigned to the normal response (NR) group (n = 18), among patients with peak serum cortisol < 18 μg/dL and peak adrenocorticotropic hormone (ACTH) increase ≥two-fold, patients without obvious background pathology were assigned to the unexplainable-HD group (n = 18), whereas patients with obvious background pathology were assigned to the explainable-HD group (n = 38), and patients with peak serum cortisol < 18 μg/dL and peak ACTH increase <two-fold were assigned to the pituitary disorder (PD) group (n = 15). Inter-group comparisons were performed based on clinical characteristics. Results In the CRH challenge test, the peak plasma ACTH levels were significantly lower in the unexplainable-HD group than in the NR group, despite more than two-fold increase compared to basal levels. The increase in serum cortisol was significantly higher in the unexplainable-HD group than in the explainable-HD and PD groups. Although patients in the unexplainable-HD group showed a clear ACTH response in the insulin tolerance test, some patients had peak serum cortisol levels of < 18 μg/dL. Furthermore, attenuated diurnal variations and low normal levels of urinary free cortisol were observed. Most patients in the unexplainable-HD group were young women with chronic fatigue. However, supplementation with oral hydrocortisone at physiological doses reduced fatigue only in some patients. Conclusions Patients with unexplainable HD diagnosed by the CRH challenge test had hypothalamic–pituitary–adrenal (HPA) axis dysfunction and some patients had mild central adrenal insufficiency. Hydrocortisone supplementation reduced fatigue only in some patients, suggesting that HPA axis dysfunction may be a physiological adaptation. Further investigation of these patients may help elucidate the pathophysiology of myalgic encephalitis/chronic fatigue syndrome.

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Section: Discussioncontrasting

confidence: 68%

Clinical characteristics of patients with unexplainable hypothalamic disorder diagnosed by the corticotropin-releasing hormone challenge test: a retrospective study

et al. 2022

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“…However, other patients did not have any discernible rise in plasma ACTH levels indicating that some patients genuinely have pituitary ACTH deficiency. There have been two previous case reports which described patients with isolated ACTH deficiency, which were attributed to CRF deficiency, on the basis of normal cortisol response following CRF administration . In contrast, other papers have reported no significant rise in cortisol or ACTH secretion .…”

Section: Discussionmentioning

confidence: 99%

Clinical features and autoimmune associations in patients presenting with Idiopathic Isolated ACTH deficiency

Hannon

Hunter

Smith

et al. 2018

Clinical Endocrinology

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“…She has been in good condition by the daily administration of hydrocortisone (10 mg) alone. Low plasma ACTH response to insulin-induced hypoglycemia and exaggerated and delayed plasma ACTH response to CRH stimulation have been considered as an indicator of hypothalamic hypopituitarism [4]. However, in this case, the ACTH response to insulin-induced hypoglycemia was normal.…”

mentioning

confidence: 81%

“…Hypoglycemic stress induced by ITT is a powerful but indirect stimulus to cortisol production via activation of hypothalamus. Therefore, low ACTH response to ITT in combination with delayed and exaggerated ACTH response to CRH suggests the presence of hypothalamic insufficiency [1][2][3][4].…”

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confidence: 99%

A Case of Hypothalamic Adrenal Insufficiency Manifested Normal ACTH Response to Insulin-Induced Hypoglycemia.

Hasegawa

Ashihara

2000

Endocr J

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Abstract.A Case reportA 65-year-old Japanese woman was admitted with persistent nausea and general malaise after recovery from common cold. She had been weary from overwork for the four months prior to the appearance of symptoms. She had never received oral administration, injection or inhalation of corticosteroids, nor did she have a history of postpartum hemorrhage, but she had experienced repeated episodes of aseptic meningitis at the age of 43, 52, 59 and 60. Her menopause occurred at age 48.Physical examination on admission revealed that she was 147.0 cm height and weighed 38.0 kg. Blood pressure was 80/52 mmHg and pulse rate was 70/min. Consciousness was clear. Several patches of depigmentation were noted on the skin of anterior chest, upper back and both forearms. The pubic hair was preserved, but the axillary hair was lost. No goiter was palpable. There was no abnormal finding in the chest or abdomen. Laboratory studies on admission disclosed hyponatremia (127 mEq/1) and normochromic normocytic anemia (Hb 10.3 g/dl).

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A Case of Adrenal Insufficiency Due to Acquired Hypothalamic CRH Deficiency.

Cited by 13 publications

References 9 publications

Clinical characteristics of patients with unexplainable hypothalamic disorder diagnosed by the corticotropin-releasing hormone challenge test: a retrospective study

Clinical characteristics of patients with unexplainable hypothalamic disorder diagnosed by the corticotropin-releasing hormone challenge test: a retrospective study

Clinical features and autoimmune associations in patients presenting with Idiopathic Isolated ACTH deficiency

A Case of Hypothalamic Adrenal Insufficiency Manifested Normal ACTH Response to Insulin-Induced Hypoglycemia.

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