Idiopathic and isolated adrenocorticotropic hormone deficiency presenting as continuous epigastric discomfort without symptoms of hypoglycemia: a case report

Abstract

Background: Isolated adrenocorticotropic hormone deficiency is one kind of hypopituitarism and is triggered by various diseases including autoimmune disorder and/or autoimmune hypophysitis. Adrenocorticotropic hormone deficiency brings out various serious symptoms such as severe hypoglycemia, hypotensive shock, and disturbance of consciousness.

Case presentation: Here we report a case of 65-year-old Japanese man who developed idiopathic and isolated adrenocorticotropic hormone deficiency. He had continued epigastric comfort without any symptom of hypoglycemia or any autoimmune abnormality. Since he continued to complain of mild epigastric discomfort and general malaise, he was misdiagnosed as having functional dyspepsia and a depression state and took medicine for them for several months. Infection markers and several antibodies which we examined were all negative. An abdominal computed tomography scan showed no mass in adrenal tissue; contrast magnetic resonance imaging of his brain showed that pituitary size was within normal range, and pituitary gland deep dyeing delay and/or deeply stained deficit were not observed. However, in a corticotropin-releasing hormone load test, response of adrenocorticotropic hormone and cortisol was poor after corticotropin-releasing hormone loading, and in growth hormone-releasing peptide 2 load test, adrenocorticotropic hormone response was poor, suggesting the presence of adrenocorticotropic hormone deficiency. Therefore, we started treatment with hydrocortisone, and his various symptoms were soon mitigated.

Conclusions: We should bear in mind the possibility of adrenocorticotropic hormone deficiency even when patients complain of epigastric discomfort or general malaise alone.

Keywords: ACTH deficiency; Case report; Epigastric discomfort; General malaise; Hypoglycemia.

Fig. 1

Various image inspections and load tests. a Rapid adrenocorticotropic hormone load test. Cortisol level is increased to over 5 μg/dL 60 minutes after the load, but the peak of cortisol is low (11 μg/dL). b Abdominal computed tomography scan. There is no mass in adrenal tissue. c Brain contrast magnetic resonance imaging. Pituitary size is within normal range, and there is no pituitary gland deep dyeing delay and no deeply stained deficit. ACTH adrenocorticotropic hormone

Fig. 2

Corticotropin-releasing hormone and growth hormone-releasing peptide 2 load test. a Corticotropin-releasing hormone load test. Response of adrenocorticotropic hormone and cortisol is poor after corticotropin-releasing hormone loading. b Growth hormone-releasing peptide 2 load test. Adrenocorticotropic hormone response is poor although growth hormone response is preserved. ACTH adrenocorticotropic hormone, CRH corticotropin-releasing hormone, GH growth hormone, GHRP2 growth hormone-releasing peptide 2

Fig. 3

Triple load test: thyrotropin-releasing hormone, growth hormone-releasing hormone, and gonadotropin-releasing hormone load. a Thyrotropin-releasing hormone load test. Thyroid-stimulating hormone and prolactin levels are increased after thyrotropin-releasing hormone loading. b Growth hormone-releasing hormone load test. Growth hormone level is increased after growth hormone-releasing hormone loading. c Gonadotropin-releasing hormone load test. Luteinizing hormone and follicle-stimulating hormone levels are increased after gonadotropin-releasing hormone loading. FSH follicle-stimulating hormone, GH growth hormone, GHRH growth hormone-releasing hormone, GnRH gonadotropin-releasing hormone, LH luteinizing hormone, PRL prolactin, TRH thyrotropin-releasing hormone, TSH thyroid-stimulating hormone

Fig. 4

Time course of various clinical parameters. a Time course of body temperature and C-reactive protein levels. After starting the treatment with hydrocortisone, body temperature and C-reactive protein levels are decreased. b Time course of sodium and eosinophil levels. After starting the treatment with hydrocortisone, sodium level is increased and eosinophil level is decreased after the treatment. BT body temperature, CRP C-reactive protein, Eo eosinophil, Na sodium