scholarly journals A Case of Transient ACTH Deficiency Associated with Polymyalgia Rheumatica

2010 ◽  
Vol 1 ◽  
pp. JCM.S6215
Author(s):  
Hitoo Nishi ◽  
Yuichi Nishi ◽  
Masato Yagita ◽  
Yoshiko Tamaru ◽  
Sadayuki Matsumoto ◽  
...  
Keyword(s):  
Hpa Axis ◽  
Polymyalgia Rheumatica ◽  
Serum Cortisol ◽  
Final Diagnosis ◽  
Pituitary Hormones ◽  
Sudden Onset ◽  
Plasma Acth ◽  
Acth Deficiency ◽  
Normal Increase ◽  
Short Time

We report a case of 79-year-old man, who was diagnosed to have transient ACTH deficiency associated with polymyalgia rheumatica (PMR). The patient presented with sudden onset bilateral shoulder pain, which was gradually aggravated. Plasma ACTH was undetectable, and both serum cortisol and urinary 17-OHCS were very low. Other pituitary hormones were normal, suggesting that hypothalamo-pituitary-adrenal (HPA) axis is selectively damaged. However, within several weeks, plasma ACTH returned to normal, and showed a normal increase response to corticotropin-releasing hormone stimulation test. These results indicated that ACTH deficiency was only transient. After hydrocortisone (10 mg/day) was administered, his symptoms became suddenly improved. Based on those results and clinical course, ie, elevated erythrocyte sedimentation rate, negative rheumatoid factor and the typical symptoms, which showed improvement to glulcocorticoid therapy, the final diagnosis was PMR, which was associated with transient ACTH deficiency. This is the first report of a case of PMR, in which the HPA axis was examined in its very acute phase. It was demonstrated that the case was associated with the transient adrenocortical hypofunction, which was recovered during a short time. It is therefore possible that PMR may show a different responsiveness of HPA axis depending on its phases.

scholarly journals Missplicing due to a synonymous, T96= exonic substitution in the T-box transcription factor TBX19 resulting in isolated ACTH deficiency

2021 ◽  
Vol 2021 ◽  
Author(s):  
Ashwini Maudhoo ◽  
Avinaash Maharaj ◽  
Federica Buonocore ◽  
Gabriel Angel Martos-Moreno ◽  
Jesús Argente ◽  
...  
Keyword(s):  
Transcription Factor ◽  
Serum Cortisol ◽  
List Type ◽  
Normal Range ◽  
Plasma Acth ◽  
Exon 2 ◽  
Isolated Acth Deficiency ◽  
Acth Deficiency ◽  
T Box

Summary Congenital isolated ACTH deficiency (IAD) is a rare condition characterised by low plasma ACTH and serum cortisol with normal production of other pituitary hormones. TBX19 (also known as TPIT) is a T-box pituitary restricted transcription factor important for POMC gene transcription and terminal differentiation of POMC-expressing cells. TBX19 gene mutations have been shown to cause neonatal-onset congenital IAD. We report a neonate of Romanian origin, who presented at 15 h of life with respiratory arrest and hypoglycaemia which recurred over the following 2 weeks. Biochemical investigations revealed IAD, with undetectable serum cortisol (cortisol < 1 μg/dL; normal range (NR): 7.8–26.2) and plasma ACTH levels within the normal range (22.1 pg/mL; NR: 4.7–48.8). He responded to hydrocortisone treatment. Patient DNA was analysed by a HaloPlex next-generation sequencing array targeting genes for adrenal insufficiency. A novel homozygous synonymous mutation p.Thr96= (Chr1:168260482; c.288G>A; rs376493164; allele frequency 1 × 10−5, no homozygous) was found in exon 2 of the TBX19 gene. The effect of this was assessed by an in vitro splicing assay, which revealed aberrant splicing of exon 2 giving rise to a mutant mRNA transcript whereas the WT vector spliced exon 2 normally. This was identified as the likely cause of IAD in the patient. The predicted protein product would be non-functional in keeping with the complete loss of cortisol production and early presentation in the patient. Learning points Synonymous variants (a nucleotide change that does not alter protein sequence) usually thought to be benign may still have detrimental effects on RNA and protein function causing disease. Hence, they should not be ignored, especially if very rare in public databases. In vitro splicing assays can be employed to characterise the consequence of intronic and exonic nucleotide gene changes that may alter splicing. Establishing a diagnosis due to a TBX19 mutation is important as it defines a condition of isolated ACTH deficiency not associated with additional pituitary deficiencies.

CO-EXISTENT PRIMARY HYPOTHYROIDISM AND ISOLATED ACTH DEFICIENCY

1976 ◽  
Vol 82 (2) ◽  
pp. 467-474 ◽  
Author(s):  
T. Yamamoto ◽  
T. Ogihara ◽  
K. Miyai ◽  
Y. Kumahara ◽  
Y. Hirata
Keyword(s):  
Growth Hormone ◽  
Elevated Serum ◽  
Pituitary Hormones ◽  
Primary Hypothyroidism ◽  
Simultaneous Occurrence ◽  
Plasma Acth ◽  
Acth Deficiency ◽  
Thyrotrophic Hormone ◽  
Serum Thyroid Hormones

ABSTRACT A 52 year old man is described in whom the simultaneous occurrence of isolated adrenocorticotrophin (ACTH) deficiency and primary hypothyroidism was documented by the determination of six anterior pituitary hormones by specific radioimmunoassays after suitable stimulations. The urinary 17-hydroxycorticosteroids excretion was persistently reduced and was increased by repeated ACTH injections. The low basal metabolic rate, reduced serum thyroid hormones and elevated serum thyrotrophic hormone were consistent with primary hypothyroidism. After resumption of euthyroidism, secretory reserves of growth hormone and ACTH were assessed. The growth hormone response to insulin-induced hypoglycaemia was normal. No rise in plasma cortisol and ACTH was observed following insulin-induced hypoglycaemia. Plasma ACTH was not augmented by iv infusion of metyrapone. The pathogenesis of two co-existent endocrine deficiencies was discussed.

scholarly journals Ambient air pollutants are associated with morning serum cortisol in overweight and obese Latino youth in Los Angeles

2021 ◽  
Vol 20 (1) ◽  
Author(s):  
C. M. Toledo-Corral ◽  
T. L. Alderete ◽  
M. M. Herting ◽  
R. Habre ◽  
A. K. Peterson ◽  
...  
Keyword(s):  
Hpa Axis ◽  
Air Pollutants ◽  
Latino Youth ◽  
Serum Cortisol ◽  
Ambient Air ◽  
Pubertal Status ◽  
Ambient Air Pollutants ◽  
Morning Cortisol ◽  
Morning Serum Cortisol ◽  
Cortisol Levels

Abstract Background Hypothalamic-pituitary-adrenal (HPA)-axis dysfunction has been associated with a variety of mental health and cardio-metabolic disorders. While causal models of HPA-axis dysregulation have been largely focused on either pre-existing health conditions or psychosocial stress factors, recent evidence suggests a possible role for central nervous system activation via air pollutants, such as nitrogen dioxide (NO2), ozone (O3) and particulate matter (PM). Therefore, in an observational study of Latino youth, we investigated if monthly ambient NO2, O3, and PM with aerodynamic diameter ≤ 2.5 (PM2.5) exposure were associated with morning serum cortisol levels. Methods In this cross-sectional study, morning serum cortisol level was assessed after a supervised overnight fast in 203 overweight and obese Latino children and adolescents (female/male: 88/115; mean age: 11.1 ± 1.7 years; pre-pubertal/pubertal/post-pubertal: 85/101/17; BMI z-score: 2.1 ± 0.4). Cumulative concentrations of NO2, O3 and PM2.5 were spatially interpolated at the residential addresses based on measurements from community monitors up to 12 months prior to testing. Single and multi-pollutant linear effects models were used to test the cumulative monthly lag effects of NO2, O3, and PM2.5 on morning serum cortisol levels after adjusting for age, sex, seasonality, social position, pubertal status, and body fat percent by DEXA. Results Single and multi-pollutant models showed that higher O3 exposure (derived from maximum 8-h exposure windows) in the prior 1–7 months was associated with higher serum morning cortisol (p < 0.05) and longer term PM2.5 exposure (4–10 months) was associated with lower serum morning cortisol levels (p < 0.05). Stratification by pubertal status showed associations in pre-pubertal children compared to pubertal and post-pubertal children. Single, but not multi-pollutant, models showed that higher NO2 over the 4–10 month exposure period associated with lower morning serum cortisol (p < 0.05). Conclusions Chronic ambient NO2, O3 and PM2.5 differentially associate with HPA-axis dysfunction, a mechanism that may serve as an explanatory pathway in the relationship between ambient air pollution and metabolic health of youth living in polluted urban environments. Further research that uncovers how ambient air pollutants may differentially contribute to HPA-axis dysfunction are warranted.

scholarly journals Endocrine and Electrolyte Balances during Periovulatory Period in Cycling Mares

Animals ◽  
2021 ◽  
Vol 11 (2) ◽  
pp. 520 ◽  
Author(s):  
Katiuska Satué ◽  
Esterina Fazio ◽  
Ana Muñoz ◽  
Pietro Medica
Keyword(s):  
Estrous Cycle ◽  
Hpa Axis ◽  
Adrenocorticotropic Hormone ◽  
Venous Blood ◽  
Competitive Elisa ◽  
Direct Relation ◽  
Plasma Acth ◽  
Blood Samples ◽  
Hypothalamic Pituitary Adrenal ◽  
Stimulation Of

In cycling females, the periovulatory period is characterized by stimulation of the hypothalamic pituitary adrenal (HPA) axis. The aim of present study was to analyze the pattern and interrelationships among adrenocorticotropic hormone (ACTH), cortisol (CORT), aldosterone (ALD) and electrolytes (sodium—Na+, potassium—K+ and chloride—Cl−) during periovulatory period in cycling mares. Venous blood samples were obtained daily from a total of 23 Purebred Spanish broodmares, aged 7.09 ± 2.5 years, from day −5 to day +5 of estrous cycle, considering day 0, the day of ovulation. Plasma ACTH was measured by a fluorescent immunoassay kit, serum CORT and ALD by means of a competitive ELISA immunoassay, and plasma Na+, K+ and Cl− were quantified by an analyzer with selective electrodes for the three ions. ACTH showed higher concentrations at day 0 compared to days −5 to −1 and +1 to +3 (p < 0.05). CORT showed higher concentrations at day 0 compared to days −5 to −2 and +1 to +5 (p < 0.05). ALD showed higher concentrations at day 0 compared to days −5 to −2 (p < 0.05) and +2 (p < 0.05). Na+ and Cl− showed higher concentrations at day 0, compared to day −5 and +5. K+ showed lower concentrations at day 0 compared to day +1 (p < 0.05). The significant correlations obtained between ACTH and CORT (r = 0.20) and between ACTH and ALD (r = 0.32) suggest that although ACTH may have an effect both on CORT and ALD, there are other very important determinants that could be considered. Hence, it is possible to presume that the pituitary adrenocortical response and ALD may be involved in the ovulatory mechanisms without a direct relation with electrolyte pattern.

scholarly journals Transient sex-related changes in the mice hypothalamo–pituitary–adrenal (HPA) axis during the acute phase of the inflammatory process

1993 ◽  
Vol 2 (2) ◽  
pp. 123-127 ◽  
Author(s):  
T. Daneva ◽  
E. Spinedi ◽  
R. Hadid ◽  
M.-C. Jacquier ◽  
M. Giacomini ◽  
...  
Keyword(s):  
Sex Difference ◽  
Acute Phase ◽  
Hpa Axis ◽  
Plasma Levels ◽  
Potential Role ◽  
Post Treatment ◽  
Male Mice ◽  
Plasma Acth ◽  
Endogenous Sex Hormones

The potential role of endogenous sex hormones in regulating hypothalamo–pituitary–adrenal (HPA) axis function was investigated after a single injection of endotoxin in adult (8 week old) BALB/c mice of both sexes. The effect of LPS on plasma ACTH, corticosterone (B), testosterone and oestradiol (E) levels and on anterior pituitary (AP) ACTH and adrenal B contents at different times after treatment was studied. The results indicate that: (a) basal B but not ACTH plasma levels were significantly higher in female than in male mice; (b) LPS significantly increased both ACTH and B plasma levels over the baseline 2 h after injection, both hormone levels being higher in female than in male mice; (c) although plasma ACTH concentrations recovered the basal value at 72 h after LPS in animals of both sexes, plasma B levels returned to the baseline only at 120 h after treatment; (d) E plasma levels significantly increased 2 h after LPS and returned to the baseline at 72 h post-treatment, in both sexes; (e) at 2 h after LPS, testosterone plasma levels significantly decreased in male mice and increased in female mice, recovering the baseline level at 120 and 72 h after LPS, respectively; (f) AP ACTH content was similar in both sexes in basal condition and it was significantly diminished 72 h post-treatment without sex difference; whereas AP ACTH returned to basal content 120 h after LPS in males, it remained significantly decreased in females; (g) basal adrenal B content was higher in female than in male mice, and it significantly increased in both sexes 2 h post-LPS, maintaining this sex difference. Whereas adrenal B returned to basal content 72 h after treatment in male mice, it remained significantly enhanced up to 120 h post-LPS in female animals. The data demonstrate the existence of a clear sexual dimorphism in basal condition and during the acute phase response as well as in the recovery of the HPA axis function shortly after infection.

scholarly journals Post-surgical hypocortisolism after removal of an adrenal incidentaloma: is it predictable by an accurate endocrinological work-up before surgery?

2010 ◽  
Vol 162 (1) ◽  
pp. 91-99 ◽  
Author(s):  
Cristina Eller-Vainicher ◽  
Valentina Morelli ◽  
Antonio Stefano Salcuni ◽  
Massimo Torlontano ◽  
Francesca Coletti ◽  
...  
Keyword(s):  
Hpa Axis ◽  
Adrenal Incidentaloma ◽  
Serum Cortisol ◽  
Urinary Free Cortisol ◽  
Tolerance Test ◽  
Surgical Removal ◽  
Acth Stimulation ◽  
Unilateral Adrenalectomy ◽  
Free Cortisol ◽  
Few Data

ObjectiveFew data are available regarding the need of steroid substitutive therapy after unilateral adrenalectomy for adrenal incidentaloma (AI). It is unknown whether, before surgery, the hypothalamic–pituitary–adrenal (HPA) axis secretion parameters can predict post-surgical hypocortisolism.AimThis study aimed to evaluate whether, in AI patients undergoing unilateral adrenalectomy, post-surgical hypocortisolism could be predicted by the parameters of HPA axis function.DesignProspective, multicenter.MethodsA total of 60 patients underwent surgical removal of AI (surgical indication: 29 subclinical hypercortisolism (SH); 31 AI dimension). Before surgery, SH was diagnosed in patients presenting at least three criteria out of urinary free cortisol (UFC) levels>60 μg/24 h, cortisol after 1-mg dexamethasone suppression test (1 mg-DST)>3.0 μg/dl, ACTH levels<10 pg/ml, midnight serum cortisol (MSC)>5.4 μg/dl.Two months after surgery, HPA axis function was assessed by low dose ACTH stimulation test or insulin tolerance test when needed: 39 patients were affected (Group B) and 21 were not affected (Group A) with hypocortisolism. The accuracy in predicting hypocortisolism of pre-surgical HPA axis parameters or their combinations was evaluated.ResultsThe presence of >2 alterations among 1 mg-DST>5.0 μg/dl, ACTH<10 pg/ml, elevated UFC and MSC has the highest odds ratio (OR) for predicting post-surgical hypocortisolism (OR 10.45, 95% confidence interval, CI 2.54–42.95, P=0.001). Post-surgical hypocortisolism was predicted with 100% probability by elevated UFC plus MSC levels, but not ruled out even in the presence of the normality of all HPA axis parameters.ConclusionPost-surgical hypocortisolism cannot be pre-surgically ruled out. A steroid substitutive therapy is indicated after unilateral adrenalectomy for SH or size of the adenoma.

scholarly journals Observations of High-Frequency Activity of EV Lacertae

1995 ◽  
Vol 151 ◽  
pp. 82-84
Author(s):  
B.E. Zhilyaev ◽  
I.A. Verlyuk
Keyword(s):  
High Frequency ◽  
Sudden Onset ◽  
X Rays ◽  
Long Duration ◽  
Cataclysmic Binaries ◽  
Wide Range ◽  
Classical Models ◽  
Low Mass ◽  
Ev Lac ◽  
Short Time

The flare star EV Lac shows a wide variety of activity phenomena on a wide range of time scales. These are the well-known “classical” solar-like flares, the short-time flare events (Gershberg & Petrov 1986, Karapetian & Zalinian 1991, Tovmassian & Zalinian 1988, Tsvetkov et al. 1986, Zhilyaev 1994), and the long-duration variations caused by starspots and rotation (Petterson 1980). However, the “classical” flares are not such a frequent phenomenon as one gets used to think. As noted by Roizman & Kabitchev (1985), a considerable fraction of flares shows a complicated multipeak structure with sudden onset and decay. Their interpretation along the lines of classical models favored for solar-like flares is very problematic.Here, we report the detection of high-frequency chaotic variability (flickering) in EV Lac. Optical flickering is observed in cataclysmic binaries, and in X-rays it is observed in low-mass X-ray binaries. As a rule, flickering is an accretion phenomenon. Strange as it may seem, EV Lac as a visual binary has the analogous property.

scholarly journals Idiopathic Isolated Acquired Steroid Dependent SO Palsy: A Rare Case Report

2019 ◽  
Vol 2019 ◽  
pp. 1-4
Author(s):  
Isha Agarwal ◽  
Mayuresh Naik ◽  
HarinderSingh Sethi
Keyword(s):  
Body Weight ◽  
Oral Prednisolone ◽  
Final Diagnosis ◽  
Sudden Onset ◽  
Step Test ◽  
Superior Oblique Palsy ◽  
Steroid Dependent ◽  
Rare Case Report ◽  
Oral Steroids ◽  
Superior Oblique

15-year-old boy presented with sudden onset, stable, nonprogressive painless diplopia (greatest in right gaze and inferior field of view) and hyperdeviation of left eye for a year. On ophthalmic examination, the patient had uncrossed diplopia with tilt and separation maximum in dextrodepression. On Park’s three step test, left eye hypertropia increased on right gaze and left tilt suggestive of left superior oblique (SO) palsy. On prism bar cover test (PBCT), deviation was more than 25 PD base-down over the left eye for both distance and near in all gazes. MRI head and orbit revealed a normal study while the myasthenia and inflammatory work-up was unremarkable. A provisional diagnosis of “Idiopathic Acquired Left Superior Oblique Palsy” was made and the patient was given trial of oral steroids at 1 mg/kg body weight. At 6 weeks, patient’s diplopia resolved and PBCT neutralised at 6PD. Oral steroids were gradually tapered off by 10 mg per week with weekly follow-up. Upon decreasing the dose of prednisolone to 5 mg, intermittent diplopia and 18 PD left hypertropia reappeared. When patient was again restarted on oral steroids at 1 mg/kg body weight, diplopia-hypertropia disappeared at 10 mg OD prednisolone only to reappear at 5 mg OD dosage, leading to the final diagnosis of a “Steroid Dependent Isolated Superior Oblique Palsy”. Presently, the patient is maintained on a daily dose of 10 mg oral prednisolone.

scholarly journals Hypoactivity of the Hypothalamo-Pituitary-Adrenocortical Axis during Recovery from Chronic Variable Stress

Endocrinology ◽  
2006 ◽  
Vol 147 (4) ◽  
pp. 2008-2017 ◽  
Author(s):  
Michelle M. Ostrander ◽  
Yvonne M. Ulrich-Lai ◽  
Dennis C. Choi ◽  
Neil M. Richtand ◽  
James P. Herman
Keyword(s):  
Mrna Expression ◽  
Chronic Stress ◽  
Hpa Axis ◽  
Male Rats ◽  
Plasma Acth ◽  
Novel Environment ◽  
Systemic Hypoxia ◽  
Delayed Recovery ◽  
Chronic Variable Stress

Chronic stress induces both functional and structural adaptations within the hypothalamo-pituitary-adrenocortical (HPA) axis, suggestive of long-term alterations in neuroendocrine reactivity to subsequent stressors. We hypothesized that prior chronic stress would produce persistent enhancement of HPA axis reactivity to novel stressors. Adult male rats were exposed to chronic variable stress (CVS) for 1 wk and allowed to recover. Plasma ACTH and corticosterone levels were measured in control or CVS rats exposed to novel psychogenic (novel environment or restraint) or systemic (hypoxia) stressors at 16 h, 4 d, 7 d, or 30 d after CVS cessation. Plasma ACTH and corticosterone responses to psychogenic stressors were attenuated at 4 d (novel environment and restraint) and 7 d (novel environment only) recovery from CVS, whereas hormonal responses to the systemic stressor were largely unaffected by CVS. CRH mRNA expression was up-regulated in the paraventricular nucleus of the hypothalamus (PVN) at 16 h after cessation of CVS, but no other alterations in PVN CRH or arginine vasopressin mRNA expression were observed. Thus, in contrast to our hypothesis, reductions of HPA axis sensitivity to psychogenic stressors manifested at delayed recovery time points after CVS. The capacity of the HPA axis to respond to a systemic stressor appeared largely intact during recovery from CVS. These data suggest that chronic stress selectively targets brain circuits responsible for integration of psychogenic stimuli, resulting in decreased HPA axis responsiveness, possibly mediated in part by transitory alterations in PVN CRH expression.

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