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Clinical Immunology (Orlando, Fla.) Mar 2024Adrenal hemorrhage (AH) can occur in patients with antiphospholipid Syndrome (APS). We aimed to characterize the clinical manifestations, treatments, and outcomes of...
BACKGROUND
Adrenal hemorrhage (AH) can occur in patients with antiphospholipid Syndrome (APS). We aimed to characterize the clinical manifestations, treatments, and outcomes of patients presenting with APS-associated AH (APS-AH) through a retrospective cohort and a systematic literature review (SLR).
METHODS
We performed a mixed-source approach combining a multicenter cohort with an SLR of patients with incident APS-AH. We included patients from Mayo Clinic and published cases with persistent positivity for antiphospholipid antibodies and presenting with AH, demonstrated by imaging or biopsy. We extracted demographics, clinical characteristics, laboratory findings, treatment strategies, and outcomes (primary adrenal insufficiency and mortality). We used Kaplan-Meier and Cox models for survival analysis.
RESULTS
We included 256 patients in total, 61 (24%) from Mayo Clinic and 195 (76%) from the SLR. The mean age was 46.8 (SD 15.2) years, and 45% were female. 69% of patients had bilateral adrenal involvement and 64% presented adrenal insufficiency. The most common symptoms at presentation were abdominal pain in 79%, and nausea and vomiting 46%. Hyponatremia (77%) was the most common electrolyte abnormality. Factors associated with primary adrenal insufficiency were bilateral adrenal involvement at initial imaging (OR 3.73, CI; 95%, 1.47-9.46) and anticardiolipin IgG positivity (OR 3.80, CI; 95%, 1.30-11.09). The survival rate at five years was 82%. History of stroke was associated with 3.6-fold increase in mortality (HR 3.62, 95% CI; 1.33-9.85).
CONCLUSION
AH is a severe manifestation of APS with increased mortality. Most patients developed permanent primary adrenal insufficiency, particularly those positive for anticardiolipin IgG and bilateral adrenal involvement.
Topics: Female; Humans; Male; Middle Aged; Addison Disease; Antiphospholipid Syndrome; Hemorrhage; Immunoglobulin G; Multicenter Studies as Topic; Retrospective Studies; Adult
PubMed: 38244823
DOI: 10.1016/j.clim.2024.109906 -
Pituitary Feb 2024Isolated adrenocorticotropic hormone deficiency (IAD) is considered to be a rare disease. Due to the nonspecific clinical presentation, precise data on the prevalence... (Review)
Review
Isolated adrenocorticotropic hormone deficiency (IAD) is considered to be a rare disease. Due to the nonspecific clinical presentation, precise data on the prevalence and incidence are lacking. In this systematic review, we aimed to analyse the clinical characteristics, association with autoimmune diseases, and management of acquired idiopathic IAD cases. A structured search was conducted after developing a search strategy combining terms for acquired (idiopathic) IAD. Articles describing an adult case with a diagnosis of ACTH deficiency using dynamic testing, no deficiency of other pituitary axes, and MRI of the brain/pituitary protocolled as normal, were included. Exclusion criteria were cases describing congenital IAD, cases with another aetiology for IAD, and articles where full text was not available. In total 42 articles were included, consisting of 85 cases of acquired idiopathic IAD. Distribution by sex was approximately equal (F:M; 47:38). Lethargy was the most common presenting symptom (38%), followed by weight loss (25%), anorexia (22%), and myalgia/arthralgia (12%). Eight cases (9.5%) presented with an Addison crisis. 31% of cases had an autoimmune disease at diagnosis of which Hashimoto hypothyroidism was the most frequent. Data about follow-up was scarce; dynamic testing was repeated in 4 cases of which 2 showed recovery of the adrenal axis. We report the largest case series of acquired idiopathic IAD to date. Our systematic review highlights the lack of a clear definition and diagnostic work-up. Based on the findings in this review a proposition is made for a flowchart to diagnose acquired idiopathic IAD.
Topics: Adult; Humans; Endocrine System Diseases; Adrenal Insufficiency; Adrenocorticotropic Hormone; Hypoglycemia; Genetic Diseases, Inborn
PubMed: 38151529
DOI: 10.1007/s11102-023-01366-9 -
Cognitive and Behavioral Neurology :... Jun 2023Given the sparse nature of acute mania or psychosis in primary adrenal insufficiency (PAI), physicians may not be aware of the association of these two entities.
BACKGROUND
Given the sparse nature of acute mania or psychosis in primary adrenal insufficiency (PAI), physicians may not be aware of the association of these two entities.
OBJECTIVE
To conduct a systematic review of the literature for the purpose of identifying all studies reporting mania and/or psychosis in individuals with PAI.
METHOD
We conducted a systematic review according to PRISMA (Preferred Reporting Items for Systematic Reviews and Meta-Analyses) guidelines using the PubMed, Embase, and Web of Science databases from June 22, 1970 to June 22, 2021, for the purpose of identifying all studies reporting instances of mania or psychosis associated with PAI.
RESULTS
We identified nine case reports featuring nine patients (M age = 43.3 years, male = 44.4%) over eight countries that fit our inclusion/exclusion criteria. Eight (89%) of the patients had experienced psychosis. Manic and/or psychotic symptom resolution was achieved in 100% of the cases, of which steroid replacement therapy was efficacious in seven (78%) cases and was sufficient in six (67%).
CONCLUSION
Acute mania and psychosis in the context of PAI is a very rare presentation of an already uncommon disease. Resolution of acute psychiatric change is reliably achieved with the correction of underlying adrenal insufficiency.
Topics: Humans; Male; Adult; Mania; Addison Disease; Psychotic Disorders
PubMed: 37026774
DOI: 10.1097/WNN.0000000000000340 -
Advances in Nutrition (Bethesda, Md.) Jan 2023Results from observational studies suggest that children and adolescents consuming ready-to-eat cereals (RTECs) have a healthier BMI and lower odds of overweight and... (Review)
Review
The Impact of Ready-to-Eat Cereal Intake on Body Weight and Body Composition in Children and Adolescents: A Systematic Review of Observational Studies and Controlled Trials.
Results from observational studies suggest that children and adolescents consuming ready-to-eat cereals (RTECs) have a healthier BMI and lower odds of overweight and obesity than consumers of other breakfasts or breakfast skippers. However, randomized controlled trials in children and adolescents are few and have been inconsistent in demonstrating a causal relationship between RTEC intake and body weight or body composition. The objective of this study was to evaluate the effect of RTEC intake on body weight and body composition outcomes in children and adolescents. Prospective cohort, cross-sectional and controlled trials in children or adolescents were included. Retrospective studies and studies in subjects with disease, other than obesity, type-2 diabetes (T2D), metabolic syndrome, or prediabetes, were excluded. A search in PubMed and CENTRAL databases yielded 25 relevant studies, which were qualitatively analyzed. Fourteen of the 20 observational studies demonstrated that children and adolescents consuming RTEC have a lower BMI, lower prevalence and odds of overweight/obesity and more favorable indicators of abdominal obesity than nonconsumers or less frequent consumers. Controlled trials were few and only one reported a loss of 0.9 kg in overweight/obese children with RTEC consumption when accompanied by nutrition education. The risk of bias was low for most studies, but six had some concerns or high risk. The results were similar with presweetened and nonpresweetened RTEC. No studies reported a positive association of RTEC intake with body weight or body composition. Although controlled trials do not show a direct effect of RTEC consumption on body weight or body composition, the preponderance of observational data supports the inclusion of RTEC as part of a healthy dietary pattern for children and adolescents. Evidence also suggests similar benefits on body weight and body composition regardless of the sugar content. Additional trials are needed to determine the causality between RTEC intake and body weight and body composition outcomes. PROSPERO REGISTRATION: CRD42022311805.
Topics: Child; Humans; Adolescent; Overweight; Edible Grain; Energy Intake; Body Mass Index; Cross-Sectional Studies; Prospective Studies; Retrospective Studies; Pediatric Obesity; Body Weight; Body Composition; Randomized Controlled Trials as Topic
PubMed: 36811587
DOI: 10.1016/j.advnut.2022.11.003 -
Endocrinologia, Diabetes Y Nutricion Dec 2022Exposure to seasonal environmental factors during gestation or early in the postnatal period could influence the development of autoimmunity, determining a seasonality...
BACKGROUND
Exposure to seasonal environmental factors during gestation or early in the postnatal period could influence the development of autoimmunity, determining a seasonality in the month of birth (MOB). There are studies evaluating this potential seasonality in patients with type 1 diabetes (T1D), autoimmune thyroid diseases (AITD), and Addison's disease (ADD), but results have been controversial.
METHODS
Systematic review according to PRISMA guidelines, using PubMed, Web of Science and WorldCat databases (2005-2020) of studies that explored the association between the seasonality of the MOB and T1D, AITD and ADD. Information on sex and age, location, methodology and internal quality, seasonal patterns, hypotheses and other factors proposed to explain seasonality were extracted. Differences in season and month of birth were further discussed.
RESULTS
The initial search retrieved 300 articles, and after further screening, 11 articles fulfilled inclusion criteria and were finally selected and reviewed. 73% found a seasonal pattern and 64% showed birth peaks in spring and/or summer. Hashimoto's thyroiditis and women exhibited a higher seasonality. Ultraviolet radiation, Vitamin D levels and viral infections were identified as influencing factors.
CONCLUSIONS
The effect of certain seasonal factors during foetal development, reflected by the seasonal differences in the MOB, could contribute to the development of endocrine autoimmune diseases in predisposed patients. Further research is needed to elucidate the mechanisms underlying the observed seasonality.
Topics: Humans; Female; Diabetes Mellitus, Type 1; Ultraviolet Rays; Autoimmune Diseases; Hashimoto Disease; Autoimmunity
PubMed: 36526353
DOI: 10.1016/j.endien.2022.11.017 -
Frontiers in Endocrinology 2022In recent years, vitamin D has become the protagonist in many studies. From cardiology to oncology the spotlight was on this vitamin. While in the past it was considered... (Review)
Review
In recent years, vitamin D has become the protagonist in many studies. From cardiology to oncology the spotlight was on this vitamin. While in the past it was considered for its important role in phospho-calcium metabolism and skeletal disorders; today by studying it better, thousands of scenarios and facets have opened up on this vitamin which is actually a hormone in all respects. There are authoritative studies that demonstrate its activity and on: carcinogenesis, inflammation, autoimmunity and endocrinopathies. Its role has been studied in type 1 and type 2 diabetes mellitus, in Hashimoto or Graves' thyroiditis and even in adrenal gland diseases. In fact, there are several studies that demonstrate the possible correlations between vitamin D and: Addison's disease, Cushing disease, hyperaldosteronism or adrenocortical tumors. Moreover, this fascinating hormone and adrenal gland even seem to be deeply connected by common genetic pathways. This review aimed to analyze the works that have tried to study the possible influence of vitamin D on adrenal diseases. In this review we analyze the works that have tried to study the possible influence of vita-min D on adrenal disease.
Topics: Humans; Vitamin D; Diabetes Mellitus, Type 2; Adrenal Glands; Hyperaldosteronism; Vitamins; Hormones
PubMed: 36313775
DOI: 10.3389/fendo.2022.1001065 -
Applied Neuropsychology. Adult 2023Addison's disease (AD) entails a chronic insufficient production of gluco- and mineralocorticoids. Fatigue and decreased quality of life are frequently reported...
Addison's disease (AD) entails a chronic insufficient production of gluco- and mineralocorticoids. Fatigue and decreased quality of life are frequently reported symptoms, but little is known about its effects on cognition. This study aims to explore the existence of cognitive impairment in patients with AD and the influence of treatment regimens. We conducted a systematic review. Inclusion criteria were met by 10 articles, most of them ranked as intermediate quality. Three studies analyzed the relationship between AD and cognitive impairment; one explored the effect of delaying treatment showing no effect on cognitive performance, and another one studied the effect of fludrocortisone treatment. Episodic memory was the most frequent cognitive domain impaired across studies, in comparison to healthy controls. Two papers investigated the relationship between impaired sleep quality and poor cognitive performance. Two studies related cognitive impairments with hypocortisolism-derived brain neuroglycopenia. Two studies investigated the effect of DHEA substitution. In conclusion, patients exhibit a moderately reduced performance in verbal learning. The pathophysiology of this impairment is likely multifactorial. Future studies should include larger sample sizes, the use of comprehensive and multi-domain neuropsychological and behavioral protocols, and neuroimaging.
PubMed: 35767730
DOI: 10.1080/23279095.2022.2090256 -
European Journal of Endocrinology Jun 2022The incidence of adrenal crisis (AC) remains high, particularly for people with primary adrenal insufficiency, despite the introduction of behavioural interventions. The...
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.
METHODS
MEDLINE, MEDLINE in Process, EMBASE, ERIC, Cochrane CENTRAL, CINAHL, PsycINFO, the Health Management Information Consortium and trial registries were searched from inception to November 2021. Three reviewers independently selected studies and extracted data. Two reviewers appraised the studies for the risk of bias.
RESULTS
Seven observational or mixed methods studies were identified where interventions were designed to prevent AC in adrenal insufficiency. Patient education was the focus of all interventions and utilised the same two behaviour change techniques, 'instruction on how to perform a behaviour' and 'pharmacological support'. Barrier and facilitator themes aiding or hindering the intervention included knowledge, behaviour, emotions, skills, social influences and environmental context and resources. Most studies did not measure effectiveness, and assessment of knowledge varied across studies. The study quality was moderate.
CONCLUSION
This is an emerging field with limited studies available. Further research is required in relation to the development and assessment of different behaviour change interventions to prevent AC.
Topics: Addison Disease; Adult; Humans; Patient Education as Topic
PubMed: 35536876
DOI: 10.1530/EJE-21-1248 -
JMIR MHealth and UHealth Mar 2022Engagement is essential for the effectiveness of digital behavior change interventions. Existing systematic reviews examining hypertension self-management interventions... (Review)
Review
BACKGROUND
Engagement is essential for the effectiveness of digital behavior change interventions. Existing systematic reviews examining hypertension self-management interventions via mobile apps have primarily focused on intervention efficacy and app usability. Engagement in the prevention or management of hypertension is largely unknown.
OBJECTIVE
This systematic review explores the definition and role of engagement in hypertension-focused mobile health (mHealth) interventions, as well as how determinants of engagement (ie, tailoring and interactivity) have been implemented.
METHODS
A systematic review of mobile app interventions for hypertension self-management targeting adults, published from 2013 to 2020, was conducted. A total of 21 studies were included in this systematic review.
RESULTS
The engagement was defined or operationalized as a microlevel concept, operationalized as interaction with the interventions (ie, frequency of engagement, time or duration of engagement with the program, and intensity of engagement). For all 3 studies that tested the relationship, increased engagement was associated with better biomedical outcomes (eg, blood pressure change). Interactivity was limited in digital behavior change interventions, as only 7 studies provided 2-way communication between users and a health care professional, and 9 studies provided 1-way communication in possible critical conditions; that is, when abnormal blood pressure values were recorded, users or health care professionals were notified. The tailoring of interventions varied at different aspects, from the tailoring of intervention content (including goals, patient education, advice and feedback from health professionals, reminders, and motivational messages) to the tailoring of intervention dose and communication mode. Tailoring was carried out in a number of ways, considering patient characteristics such as goals, preferences, disease characteristics (eg, hypertension stage and medication list), disease self-management experience levels, medication adherence rate, and values and beliefs.
CONCLUSIONS
Available studies support the importance of engagement in intervention effectiveness as well as the essential roles of patient factors in tailoring, interactivity, and engagement. A patient-centered engagement framework for hypertension self-management using mHealth technology is proposed here, with the intent of facilitating intervention design and disease self-management using mHealth technology.
Topics: Adult; Biomedical Technology; Humans; Hypertension; Mobile Applications; Self-Management; Telemedicine
PubMed: 35234655
DOI: 10.2196/29415 -
World Neurosurgery Apr 2022The aim of this study was to assess rates of adjacent segment degeneration (ASDeg), adjacent segment disease (ASDis), and reoperation rates as a result of adjacent... (Meta-Analysis)
Meta-Analysis
The Incidence of Adjacent Segment Pathology After Cervical Disc Arthroplasty Compared with Anterior Cervical Discectomy and Fusion: A Systematic Review and Meta-Analysis of Randomized Clinical Trials.
OBJECTIVE
The aim of this study was to assess rates of adjacent segment degeneration (ASDeg), adjacent segment disease (ASDis), and reoperation rates as a result of adjacent segment pathology in patients who have undergone anterior cervical discectomy and fusion (ACDF) versus cervical disc arthroplasty (CDA).
METHODS
A comprehensive search of randomized controlled trials was performed in PubMed from 2012 to 2019. Relevant studies included were assessed for quality using the Cochrane Beck Review Group guidelines. Rates of ASDeg, ASDis, and reoperation because of adjacent segment pathology were extracted and included in the final analysis. A random-effects and fixed-effects model was run among studies that showed high and low heterogeneity, respectively.
RESULTS
A total of 19 studies were included in the final analysis, comprising 4655 patients. Overall, ACDFs reported significantly higher rates of ASDeg (19.7% vs. 14.4%; P < 0.001), ASDis (6.1% vs. 3.8%; P < 0.001), and reoperation rates (6.1% vs. 3.1%; P < 0.001) compared with CDAs, which was further corroborated in fixed-effects analysis. When stratified by length of follow-up, a significant difference was seen in ASDeg, ASDis, and reoperation rates of studies with follow-up of 12-48 months versus >48 months, with the exception of ASDeg rates in the CDA cohort.
CONCLUSIONS
Our study shows CDA results in significantly lower ASDeg, ASDis, and reoperation rates. Although CDA may be a viable alternative to ACDF, further long-term studies are warranted to ensure consistency and establish longevity of our findings.
Topics: Arthroplasty; Cervical Vertebrae; Diskectomy; Humans; Incidence; Intervertebral Disc Degeneration; Randomized Controlled Trials as Topic; Spinal Fusion
PubMed: 35085804
DOI: 10.1016/j.wneu.2022.01.072