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Epilepsia 2022 Jun 27. Epub 2022 Jun 27.

Mother-Child Center - Quebec City University Hospital, QC, Canada.

Objective: Hormonal therapy is a standard treatment for children with infantile spasms. However, the high doses given, and long treatment duration expose patients to the risk of adrenal insufficiency (AI). This study aims to quantify the cumulative incidence of AI among children with infantile spasms treated with high-dose corticosteroids and/or adrenocorticotropic hormone. Read More

J Immunother Cancer 2022 Jun;10(6)

Department of Medicine, University of Chicago, Chicago, Illinois, USA

Background: Patients with cancer were excluded from phase 3 COVID-19 vaccine trials, and the immunogenicity and side effect profiles of these vaccines in this population is not well understood. Patients with cancer can be immunocompromised from chemotherapy, corticosteroids, or the cancer itself, which may affect cellular and/or humoral responses to vaccination. PD-1 is expressed on T effector cells, T follicular helper cells and B cells, leading us to hypothesize that anti-PD-1 immunotherapies may augment antibody or T cell generation after vaccination. Read More

J Neuroendocrinol 2022 Jun 3. Epub 2022 Jun 3.

Department of Endocrinology, University Hospital Center Zagreb, Zagreb, Croatia.

After successful treatment for Cushing's syndrome (CS), secondary adrenal insufficiency develops as a result of the prior suppression of the hypothalamic-pituitary-adrenal (HPA) axis by excess cortisol in the body. Until the recovery of the HPA axis, glucocorticoid replacement therapy is required to enable normal functioning of the body and prevent adrenal crisis. Significant variation in the median time of recovery of the HPA axis is found in various cohorts of CS patients ranging from several weeks to years. Read More

Front Endocrinol (Lausanne) 2022 1;13:877341. Epub 2022 Jun 1.

Department of Cardiology, State Key Laboratory of Complex Severe and Rare Diseases, Peking Union Medical College Hospital, Chinese Academy of Medical Science and Peking Union Medical College, Beijing, China.

Background: Catecholamine excess arising from pheochromocytomas and paragangliomas (PPGLs) can cause a wide spectrum of cardiac manifestations. Although there are reviews of reported cases, these reviews lack detailed data, which makes it impossible to perform an accurate analysis. In this study, we conducted a comprehensive analysis of cardiovascular complications (CCs), including PPGL-related myocardial injury, cardiogenic shock, and arrhythmias requiring antiarrhythmic therapy, in a large cohort of patients with PPGL. Read More

World J Surg Oncol 2022 Jun 8;20(1):185. Epub 2022 Jun 8.

Department of Gastroenterological Surgery, Saitama Cancer Center, Saitama, 362-0806, Japan.

Background: Paraganglioma of the urinary bladder (Pub) is rare and presents with clinical symptoms caused by catecholamine production and release. The typical symptoms of Pub are hypertension, macroscopic hematuria, and a hypertensive crisis during micturition. The average size of detected Pubs is approximately 3 cm. Read More

Indian J Endocrinol Metab 2022 Jan-Feb;26(1):50-54. Epub 2022 Apr 27.

Department of Endocrinology, Madurai Medical College, Madurai, Tamil Nadu, India.

Aims And Objectives: Clinical, biochemical, and radiological profiles of Addison's disease and to assess the various etiological spectrum of primary adrenal insufficiency (PAI) in adults.

Materials And Methods: A retrospective cohort study was carried out in the Department of Endocrinology, Madurai Medical College, Madurai between January 2014 and January 2021 over a 7-year period.

Inclusion Criteria: All the patients with clinical symptoms and or signs of suspected PAI, such as hyperpigmentation, weight loss, persistent nausea or vomiting, fatigue, and hypotension, were recruited. Read More

BMJ Case Rep 2022 Jun 1;15(6). Epub 2022 Jun 1.

Growth and Endocrine Unit, Hirabai Cowasji Jehangir Medical Research Institute, Pune, Maharashtra, India

We present a case of a young girl who presented with hypertensive crisis and recent onset weight gain with hirsutism. On evaluation for Cushing syndrome (CS), her cortisol concentration was high, showed a paradoxical cortisol rise on dexamethasone suppression and the adrenocorticotropic hormone (ACTH) was low. Adrenal imaging showed normal adrenal morphology. Read More

J Clin Res Pediatr Endocrinol 2022 May 31. Epub 2022 May 31.

Department of Pediatric and Adolescent Endocrinology, Pediatric Institute, Jagiellonian University - Medical College, Cracow, Poland.

Objective: Oncologic treatment can affect the adrenal glands, which in stressful situations may lead to life threatening adrenal crisis. The aim of the study was to assess adrenal function in pediatric acute lymphoblastic leukemia (ALL) survivors and to find best markers for this assessment.

Methods: Forty-three ALL survivors, mean age 8. Read More

Genes (Basel) 2022 Apr 20;13(5). Epub 2022 Apr 20.

Faculty of Medicine, University of Ljubljana, Vrazov Trg 2, SI-1000 Ljubljana, Slovenia.

Nicotinamide nucleotide transhydrogenase (NNT) deficiency causes primary adrenal insufficiency (PAI) and possibly some extra-adrenal manifestations. A limited number of these patients were previously described. We present the clinical and genetic characteristics of three family members with a biallelic novel pathogenic variant in the gene. Read More

Crit Care Nurs Q 2022 Jul-Sep 01;45(3):266-284

Division of Pulmonology and Critical Care, Allegheny Health Network, Pittsburgh, Pennsylvania (Drs Nasrullah, Azharuddin, Young, and Dumont); and Department of Nursing, Allegheny Health Network, Pittsburgh, Pennsylvania (Ms Kejas).

Endocrine emergencies are underdiagnosed and often overlooked amid the management of severe multisystem pathologies in critically ill patients in the medical intensive care unit (ICU). In an appropriate clinical scenario, a low threshold of suspicion should be kept to investigate for various life-threatening, yet completely treatable, endocrinopathies. Prompt identification and treatment of endocrine emergencies such as diabetic ketoacidosis, myxedema coma, thyroid storm, and/or adrenal insufficiency leads to fewer complications, shorter ICU and hospital stay, and improved survival. Read More

Horm Res Paediatr 2022 May 24. Epub 2022 May 24.

Introduction: There is emerging speculation that the inflammatory state associated with SARS-CoV-2 infection may trigger autoimmune conditions, but no causal link is established. There are reports of autoimmune thyroiditis, and adrenal insufficiency in adults post-COVID-19. We describe the first pediatric report of autoimmune adrenal insufficiency and hypothyroidism after COVID-19. Read More

Urol Case Rep 2022 Jul 7;43:102108. Epub 2022 May 7.

Endocrinology Department, Academic Hospital Hedi Chaker, Sfax, Tunisia.

Ectopic pheochromocytomas, also called paragangliomas, are defined as catecholamine -secreting tumors, which develop outside the adrenal medulla. Pheochromocytomas of the urinary tract represent less than 1% of all paragangliomas and are most commonly located in the bladder. Nevertheless, prostatic pheochromocytoma is an extremely rare clinical entity and only a few cases have been reported in the medical literature. Read More

Surgery 2022 May 16. Epub 2022 May 16.

Department of Digestive, Hepato-biliary and Endocrine Surgery, Referral Center for Rare Adrenal Diseases, Cochin Hospital, APHP, Paris, France; Department of Hepato-Biliary and Pancreatic Surgery and Liver Transplantation, AP-HP Pitié-Salpêtrière Hospital, Paris, France; Department of General, Visceral, and Endocrine Surgery, Pitié-Salpêtrière Hospital, AP-HP, Paris, France; Sorbonne University, Paris, France. Electronic address:

Background: Pheochromocytomas and paragangliomas can induce severe cardiovascular manifestations such as Takotsubo-like cardiomyopathy. What the perioperative outcomes are of patients presenting with pheochromocytomas/paragangliomas preceded by Takotsubo-like cardiomyopathy remains an unresolved question.

Methods: From 2006 to 2019, all patients who underwent surgery for pheochromocytomas/paragangliomas preceded by Takotsubo-like cardiomyopathy were included from 3 high-volume centers, with specific attention to perioperative hemodynamic instability and postoperative outcomes. Read More

Endocrine 2022 Jun 18;77(1):1-10. Epub 2022 May 18.

Department of Endocrinology, Karolinska University Hospital, SE-17176, Stockholm, Sweden.

Purpose: Review the literature concerning adrenal insufficiency (AI) and adrenal crisis (AC) in adolescents and young adults.

Methods: Searches of PubMed identifying relevant reports up to March 2022.

Results: AI is rare disorder that requires lifelong glucocorticoid replacement therapy and is associated with substantial morbidity and occasional mortality among adolescents and young adults. Read More

Horm Res Paediatr 2022 May 13. Epub 2022 May 13.

Introduction There is wide variation in reported rates of acute adrenal insufficiency (AI) related adverse events (sick day episodes and adrenal crises) between centres. This study aimed to evaluate the level of consensus on criteria considered essential for defining and managing these events in children with Congenital Adrenal Hyperplasia. Methods Active users of the International Congenital Adrenal Hyperplasia & International Disorders of Sex Development (I-CAH/I-DSD) Registries (n=66), non-active users of I-CAH/I-DSD (n=35) and the EuRRECa e-Reporting Registry (n=10) were approached to complete an online survey. Read More

Best Pract Res Clin Endocrinol Metab 2022 05 6;36(3):101668. Epub 2022 May 6.

Kobe University School of Medicine, 7-5-1, Kusunoki-cho, Chuo-ku, Kobe, 650-0017, Japan; Department of Diabetes and Endocrinology, Nara Medical University, 840 Shijo-cho, Kashihara, Nara, 634-8522, Japan.

Immune checkpoint inhibitors (ICIs) have been recently proposed as a strategy for treating anti-malignant neoplasms. However, this treatment leads to immune-related adverse events (irAEs) such as autoimmune endocrinopathy. Early diagnosis and appropriate treatment of ICI-related hypophysitis are essential as it can manifest as a life-threatening condition due to an adrenal crisis. Read More

Acute Crit Care 2022 May 11. Epub 2022 May 11.

Regional Cardiovascular Disease Center, Chungbuk National University Hospital, Cheongju, Korea.

The main mechanism of Takotsubo cardiomyopathy (TCM) is catecholamine-induced acute myocardial stunning. Pheochromocytoma, a catecholamine-secreting tumor, can cause several cardiovascular complications, including hypertensive crisis, myocardial infarction, toxic myocarditis, and TCM. A 29-year-old woman presented to our hospital with general weakness, vomiting, dyspnea, and chest pain. Read More

Eur J Endocrinol 2022 Jun 1;187(1):S1-S20. Epub 2022 Jun 1.

School of Nursing, Institute of Clinical Sciences.

Objective: The incidence of adrenal crisis (AC) remains high, particularly for people with primary adrenal insufficiency, despite the introduction of behavioural interventions. The present study aimed to identify and evaluate available evidence of interventions aiming to prevent AC in primary adrenal insufficiency.

Design: This study is a systematic review of the literature and theoretical mapping. Read More

J Clin Pathol 2022 May 9. Epub 2022 May 9.

Clinical Biochemistry, Bedfordshire Hospitals NHS Foundation Trust, Bedford, UK.

Adrenal insufficiency (AI), first described by Thomas Addison in 1855, is characterised by inadequate hormonal production by the adrenal gland, which could either be primary, due to destruction of the adrenal cortex, or secondary/tertiary, due to lack of adrenocorticotropic hormone or its stimulation by corticotropin-releasing hormone. This was an invariably fatal condition in Addison's days with most patients dying within a few years of diagnosis. However, discovery of cortisone in the 1940s not only improved the life expectancy of these patients but also had a dramatic effect on their overall quality of life. Read More

Front Endocrinol (Lausanne) 2022 20;13:849188. Epub 2022 Apr 20.

Department of Endocrinology, University Medical Center Groningen, University of Groningen, Groningen, Netherlands.

Objective: To compare cortisol pharmacokinetics and pharmacodynamics mapped through several glucocorticoid sensitive pathways in patients on hydrocortisone substitution with or without an adrenal crisis.

Design: A analysis of a previously conducted randomized controlled trial in patients with secondary adrenal insufficiency examining the effects of 2 weight-adjusted hydrocortisone doses.

Methods: Comparisons were primarily made on a hydrocortisone dose of 0. Read More

Canine Med Genet 2022 May 3;9(1). Epub 2022 May 3.

University College Dublin Veterinary Hospital, University College Dublin, Dublin, Ireland.

Hypoadrenocorticism is characterized by a reduction in mineralocorticoid and/or glucocorticoid production by the adrenal glands. Several subtypes have been described with different clinical and clinicopathological consequences. Most affected dogs have vague and non-specific signs that precede an eventual life-threatening crisis. Read More

AACE Clin Case Rep 2022 Apr 25. Epub 2022 Apr 25.

Division of Endocrinology-Diabetes and Metabolism, Saint Barnabas Medical Center, 94 Old Short Hills Road, Livingston 07039, New Jersey.

Background/objective: Adrenal crisis (AC) is an acute life-threatening condition that can occur in patients with primary or secondary adrenal insufficiency who are already on glucocorticoid replacement therapy or can be a first presentation of adrenal insufficiency. Vaccination with Tdap, influenza and pneumococcal vaccine has been reported as a cause of AC. Our objective is to present a case of AC precipitated by COVID-19 mRNA vaccination in patient with hypopituitarism. Read More

J Assoc Physicians India 2022 Apr;70(4):11-12

Mandya Institute of Medical Sciences, Mandya, Karnataka.

Tuberculosis may lead to adrenal insufficiency by direct glandular involvement, by extra glandular infection or as a by-product of Anti tuberculous therapy. Subclinical adrenal insufficiency becomes important in stressful situation which require increased release of ACTH to meet heightened metabolic demands. Our objective was to determine the adrenal reserve in patients with pulmonary tuberculosis and to determine the effect of ATT on adrenal function. Read More

Eur Heart J Case Rep 2022 Mar 7;6(3):ytac098. Epub 2022 Mar 7.

Department of Cardiology, Institute for Clinical and Experimental Medicine, Vídeňská 1958/9, Prague, Czech Republic.

Background: Pheochromocytoma is a neuroendocrine tumour originating from the chromaffin cells of adrenal glands or sympathetic paraganglia. It produces most frequently norepinephrine, epinephrine, and dopamine. As a result of non-specific and variable clinical presentation, pheochromocytoma is difficult to diagnose. Read More

J Clin Endocrinol Metab 2022 Jun;107(7):2057-2064

Clinical Division and Laboratory of Intensive Care Medicine, Department of Cellular and Molecular Medicine, KU Leuven University, B-3000 Leuven, Belgium.

Critical illnesses are hallmarked by increased systemic cortisol availability, a vital part of the stress response. Acute stress may trigger a life-threatening adrenal crisis when a disease of the hypothalamic-pituitary-adrenal (HPA) axis is present and not adequately treated with stress doses of hydrocortisone. Stress doses of hydrocortisone are also used to reduce high vasopressor need in patients suffering from septic shock, in the absence of adrenal insufficiency. Read More

mBio 2022 04 31;13(2):e0047322. Epub 2022 Mar 31.

Department of Infectious Diseases, Infection Control and Employee Health, The University of Texas MD Anderson Cancer Centergrid.240145.6, Houston, Texas, USA.

The exact cause of the disproportionate increase in COVID-19-associated mucormycosis (CAM) cases in India remains unknown. Most researchers consider the major cause of India's CAM epidemic to be the conjunction of the COVID-19 pandemic and associated corticosteroid treatment with the enormous number of Indians with diabetes mellitus (DM). However, excess CAM cases were not seen to the same extent in the Western world, where diabetes is prevalent and corticosteroids are also used extensively for COVID-19 treatment. Read More

BMJ Case Rep 2022 Mar 29;15(3). Epub 2022 Mar 29.

Endocrinology Department, Hospital de Santa Maria, Lisboa, Portugal

Pheochromocytoma and paraganglioma (PGL) are rare neuroendocrine tumours, frequently associated with genetic syndromes. We report the case of a man in his 40s with a left anterior neck mass and a history of hypertensive crisis, heavy sweating and constipation. Biochemical tests showed increased plasma and urine normetanephrines. Read More

Abdom Radiol (NY) 2022 Mar 28. Epub 2022 Mar 28.

Department of Radiology, University of Florida College of Medicine, Jacksonville, FL, USA.

Bladder pheochromocytomas (PCCs) are rare tumors that account for 0.06% of all bladder tumors and makeup 1% of all PCCs. Most PCCs are functional, and they secrete catecholamines that lead to clinical symptoms such as paroxysmal hypertension, headaches, palpitations, and sweating. Read More

Int J Neonatal Screen 2022 Mar 2;8(1). Epub 2022 Mar 2.

Division of Pediatric Endocrinology and Diabetes, The Children's Hospital at Montefiore, Albert Einstein College of Medicine, Bronx, NY 10467, USA.

Adrenoleukodystrophy (ALD) is a peroxisomal disorder affecting the nervous system, adrenal cortical function, and testicular function. Newborn screening for ALD has the potential to identify patients at high risk for life-threatening adrenal crisis and cerebral ALD. The current understanding of the natural history of endocrine dysfunction is limited. Read More

Endocrinol Diabetes Metab Case Rep 2022 Mar 1;2022. Epub 2022 Mar 1.

Department of Cardiac Sciences, University of Calgary, Calgary, Alberta, Canada.

Summary: An adrenocorticotropic hormone (ACTH)-producing pheochromocytoma (PCC)/paraganglioma is the cause of ectopic Cushing's syndrome (CS) in 5.2% of cases reported in the literature. We present a previously healthy 43-year-old woman admitted to our hospital with cushingoid features and hypertensive urgency (blood pressure = 200/120 mmHg). Read More