Pub Date : 2022-07-01Epub Date: 2022-04-23DOI: 10.1007/s40618-021-01661-x
R Pivonello, C Pivonello, C Simeoli, M C De Martino, A Colao
Cushing's Syndrome (CS), or chronic endogenous hypercortisolism, is a rare and serious disease due to corticotroph pituitary (Cushing's disease, CD) and extra-pituitary (ectopic CS) tumours overproducing ACTH, or cortisol-secreting adrenal tumours or lesions (adrenal CS). The first-line treatment for CS is represented by the surgical removal of the responsible tumour, but surgery might be unfeasible or ineffective and medical treatment can be required in a relevant percentage of patients with CS, especially CD and ectopic CS. Corticotroph pituitary and extra-pituitary tumours, as well as adrenal tumours and lesions responsible for CS express dopamine receptors (DRs), which have been found to mediate inhibition of hormone secretion and/or cell proliferation in experimental setting, suggesting that dopaminergic system, particularly DRs, might represent a target for the treatment of CS. Dopamine agonists (DAs), particularly cabergoline (CAB), are currently used as off-label treatment for CD, the most common form of CS, demonstrating efficacy in controlling hormone secretion and tumour growth in a relevant number of cases, with the improvement of clinical picture, and displaying good safety profile. Therefore, CAB may be considered a reasonable alternative treatment for persistent or recurrent CD after pituitary surgery failure, but occasionally also before pituitary surgery, as adjuvant treatment, or even instead of pituitary surgery as first-line treatment in case of surgery contraindications or refusal. A certain beneficial effect of CAB has been also reported in ectopic CS. However, the role of DAs in the clinical management of the different types of CS requires further evaluations.
{"title":"The dopaminergic control of Cushing's syndrome.","authors":"R Pivonello, C Pivonello, C Simeoli, M C De Martino, A Colao","doi":"10.1007/s40618-021-01661-x","DOIUrl":"10.1007/s40618-021-01661-x","url":null,"abstract":"<p><p>Cushing's Syndrome (CS), or chronic endogenous hypercortisolism, is a rare and serious disease due to corticotroph pituitary (Cushing's disease, CD) and extra-pituitary (ectopic CS) tumours overproducing ACTH, or cortisol-secreting adrenal tumours or lesions (adrenal CS). The first-line treatment for CS is represented by the surgical removal of the responsible tumour, but surgery might be unfeasible or ineffective and medical treatment can be required in a relevant percentage of patients with CS, especially CD and ectopic CS. Corticotroph pituitary and extra-pituitary tumours, as well as adrenal tumours and lesions responsible for CS express dopamine receptors (DRs), which have been found to mediate inhibition of hormone secretion and/or cell proliferation in experimental setting, suggesting that dopaminergic system, particularly DRs, might represent a target for the treatment of CS. Dopamine agonists (DAs), particularly cabergoline (CAB), are currently used as off-label treatment for CD, the most common form of CS, demonstrating efficacy in controlling hormone secretion and tumour growth in a relevant number of cases, with the improvement of clinical picture, and displaying good safety profile. Therefore, CAB may be considered a reasonable alternative treatment for persistent or recurrent CD after pituitary surgery failure, but occasionally also before pituitary surgery, as adjuvant treatment, or even instead of pituitary surgery as first-line treatment in case of surgery contraindications or refusal. A certain beneficial effect of CAB has been also reported in ectopic CS. However, the role of DAs in the clinical management of the different types of CS requires further evaluations.</p>","PeriodicalId":15651,"journal":{"name":"Journal of Endocrinological Investigation","volume":"45 1","pages":"1297-1315"},"PeriodicalIF":3.9,"publicationDate":"2022-07-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9184412/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"46614455","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":2,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2022-07-01Epub Date: 2021-08-24DOI: 10.1007/s40618-021-01658-6
E Martino, L Macrì, F Trimarchi
{"title":"Goiter in the models of Pre-Raphaelite Brotherhood.","authors":"E Martino, L Macrì, F Trimarchi","doi":"10.1007/s40618-021-01658-6","DOIUrl":"https://doi.org/10.1007/s40618-021-01658-6","url":null,"abstract":"","PeriodicalId":15651,"journal":{"name":"Journal of Endocrinological Investigation","volume":"45 7","pages":"1461-1462"},"PeriodicalIF":5.4,"publicationDate":"2022-07-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://sci-hub-pdf.com/10.1007/s40618-021-01658-6","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"39339528","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":2,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2022-07-01Epub Date: 2021-09-02DOI: 10.1007/s40618-021-01667-5
M E Paladino, M A Riva
In the partition of the church of San Bernardino in Caravaggio (Lombardy), Fermo Stella (c. 1490-1562) depicted a cycle of frescoes of "The Passion of Christ". In the central panel, "The Crucifixion" one of the tortures of Christ, the sponge-bearer, shows a large goiter and other signs of hypothyroidism (macroglossia, expressionless face, and puffiness around the eyes), confirming that the Renaissance artists often represented sinful and wicked individuals as affected by thyroid diseases.
{"title":"The sponge-bearer suffers from goiter: \"The Passion of Christ\" (c. 1530) by Fermo Stella.","authors":"M E Paladino, M A Riva","doi":"10.1007/s40618-021-01667-5","DOIUrl":"https://doi.org/10.1007/s40618-021-01667-5","url":null,"abstract":"<p><p>In the partition of the church of San Bernardino in Caravaggio (Lombardy), Fermo Stella (c. 1490-1562) depicted a cycle of frescoes of \"The Passion of Christ\". In the central panel, \"The Crucifixion\" one of the tortures of Christ, the sponge-bearer, shows a large goiter and other signs of hypothyroidism (macroglossia, expressionless face, and puffiness around the eyes), confirming that the Renaissance artists often represented sinful and wicked individuals as affected by thyroid diseases.</p>","PeriodicalId":15651,"journal":{"name":"Journal of Endocrinological Investigation","volume":"45 7","pages":"1463-1464"},"PeriodicalIF":5.4,"publicationDate":"2022-07-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"39379859","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":2,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2022-07-01Epub Date: 2022-02-19DOI: 10.1007/s40618-022-01767-w
G Yorulmaz, M Sahin Tekin
Purpose: With coronavirus disease 2019 (COVID-19), subacute thyroiditis (SAT) cases are on the rise all over the world. COVID-19 vaccine-associated SAT cases have also been reported. In this article, we present our data on 11 vaccine-associated SAT cases.
Methods: Eleven patients were included in the study. Type of the vaccines patients received, time to the occurrence of SAT after vaccination, symptoms and laboratory findings, treatment given, and response to treatment were evaluated.
Results: The age of patients ranged from 26 to 73. Four of the patients were males, and seven were females. Symptoms of six patients were seen after BNT162b2 Pfizer/BioNTech COVID-19 mRNA vaccine®, and four of them after Coronavac inactivated SARS-CoV-2 vaccine®. In one patient, SAT developed after the first dose of BNT162b2, administered after two doses of Coronavac. The average time to the onset of symptoms was 22 days (15-37) after vaccination.
Conclusions: The fact that both whole virus containing and genetic material containing vaccines cause SAT suggests that the trigger may be viral proteins rather than the whole viral particle. Although corticosteroids are commonly preferred in published vaccine-associated SAT cases, we preferred nonsteroidal anti-inflammatory therapy in our patients for sufficient vaccine antibody response. There is not enough information about whether patients who develop SAT can be revaccinated safely considering the ongoing pandemic. Further research is needed for a conclusion in the treatment and revaccination of these patients.
{"title":"SARS-CoV-2 vaccine-associated subacute thyroiditis.","authors":"G Yorulmaz, M Sahin Tekin","doi":"10.1007/s40618-022-01767-w","DOIUrl":"https://doi.org/10.1007/s40618-022-01767-w","url":null,"abstract":"<p><strong>Purpose: </strong>With coronavirus disease 2019 (COVID-19), subacute thyroiditis (SAT) cases are on the rise all over the world. COVID-19 vaccine-associated SAT cases have also been reported. In this article, we present our data on 11 vaccine-associated SAT cases.</p><p><strong>Methods: </strong>Eleven patients were included in the study. Type of the vaccines patients received, time to the occurrence of SAT after vaccination, symptoms and laboratory findings, treatment given, and response to treatment were evaluated.</p><p><strong>Results: </strong>The age of patients ranged from 26 to 73. Four of the patients were males, and seven were females. Symptoms of six patients were seen after BNT162b2 Pfizer/BioNTech COVID-19 mRNA vaccine®, and four of them after Coronavac inactivated SARS-CoV-2 vaccine®. In one patient, SAT developed after the first dose of BNT162b2, administered after two doses of Coronavac. The average time to the onset of symptoms was 22 days (15-37) after vaccination.</p><p><strong>Conclusions: </strong>The fact that both whole virus containing and genetic material containing vaccines cause SAT suggests that the trigger may be viral proteins rather than the whole viral particle. Although corticosteroids are commonly preferred in published vaccine-associated SAT cases, we preferred nonsteroidal anti-inflammatory therapy in our patients for sufficient vaccine antibody response. There is not enough information about whether patients who develop SAT can be revaccinated safely considering the ongoing pandemic. Further research is needed for a conclusion in the treatment and revaccination of these patients.</p>","PeriodicalId":15651,"journal":{"name":"Journal of Endocrinological Investigation","volume":"45 7","pages":"1341-1347"},"PeriodicalIF":5.4,"publicationDate":"2022-07-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8857746/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"39645224","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":2,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2022-07-01Epub Date: 2022-02-11DOI: 10.1007/s40618-022-01755-0
F Bellinato, P Gisondi, A Mantovani, G Girolomoni, G Targher
Purpose: Chronic plaque psoriasis is associated with the presence of non-alcoholic fatty liver disease (NAFLD), but the magnitude of this association remains currently uncertain. We aimed to investigate the magnitude of the association between psoriasis and the risk of prevalent and incident NAFLD, and to assess whether psoriasis severity and/or psoriatic arthritis are associated with a greater risk of NAFLD.
Methods: A systematic review and meta-analysis of observational studies evaluating the association between psoriasis and NAFLD, as diagnosed by imaging or International Classification of Diseases codes was performed. Literature search on PubMed, Scopus and Web of Science on May 3, 2021 was undertaken. Studies using liver biopsy were not available. For the meta-analysis, the random-effects modelling was adopted.
Results: We identified 15 observational (case-control and cross-sectional) studies for a total of 249,933 patients with psoriasis (49% with NAFLD) and 1,491,402 controls (36% with NAFLD). Psoriasis was associated with prevalent NAFLD (n = 11 studies; pooled random-effects odds ratio [OR] 1.96, 95% CI 1.70-2.26; I2 = 97%, p < 0.01). Psoriatic patients with NAFLD had a higher mean psoriasis area and severity index (PASI) than their counterparts without NAFLD (n = 8 studies, pooled weighted mean difference: 3.93, 95% CI 2.01-5.84; I2 = 88%, p < 0.01). The risk of NAFLD was marginally higher in patients with psoriatic arthritis than in those with psoriasis alone (n = 5 studies, pooled random-effects OR 1.83, 95% CI 0.98-3.43; I2 = 64%, p = 0.03). Sensitivity analyses did not alter these findings. Funnel plot did not show any significant publication bias. A major limitation of the study was the high degree of heterogeneity across studies.
Conclusion: Psoriasis is associated with prevalent NAFLD and this risk parallels the severity of psoriasis.
目的:慢性斑块型银屑病与非酒精性脂肪性肝病(NAFLD)相关,但这种关联的程度目前仍不确定。我们的目的是调查银屑病与NAFLD流行和发病风险之间的关联程度,并评估银屑病严重程度和/或银屑病关节炎是否与NAFLD的高风险相关。方法:对观察性研究进行系统回顾和荟萃分析,评估牛皮癣和NAFLD之间的关联,通过影像学或国际疾病分类代码进行诊断。于2021年5月3日在PubMed、Scopus和Web of Science进行文献检索。没有肝活检的研究。meta分析采用随机效应模型。结果:我们确定了15项观察性(病例对照和横断面)研究,共纳入249933例牛皮癣患者(49%患有NAFLD)和1491402例对照(36%患有NAFLD)。牛皮癣与NAFLD的流行相关(n = 11项研究;合并随机效应优势比[OR] 1.96, 95% CI 1.70-2.26;I2 = 97%, p2 = 88%, p2 = 64%, p = 0.03)。敏感性分析没有改变这些发现。漏斗图未显示任何显著的发表偏倚。该研究的一个主要局限性是研究之间的高度异质性。结论:牛皮癣与NAFLD的流行有关,这种风险与牛皮癣的严重程度相当。
{"title":"Risk of non-alcoholic fatty liver disease in patients with chronic plaque psoriasis: an updated systematic review and meta-analysis of observational studies.","authors":"F Bellinato, P Gisondi, A Mantovani, G Girolomoni, G Targher","doi":"10.1007/s40618-022-01755-0","DOIUrl":"https://doi.org/10.1007/s40618-022-01755-0","url":null,"abstract":"<p><strong>Purpose: </strong>Chronic plaque psoriasis is associated with the presence of non-alcoholic fatty liver disease (NAFLD), but the magnitude of this association remains currently uncertain. We aimed to investigate the magnitude of the association between psoriasis and the risk of prevalent and incident NAFLD, and to assess whether psoriasis severity and/or psoriatic arthritis are associated with a greater risk of NAFLD.</p><p><strong>Methods: </strong>A systematic review and meta-analysis of observational studies evaluating the association between psoriasis and NAFLD, as diagnosed by imaging or International Classification of Diseases codes was performed. Literature search on PubMed, Scopus and Web of Science on May 3, 2021 was undertaken. Studies using liver biopsy were not available. For the meta-analysis, the random-effects modelling was adopted.</p><p><strong>Results: </strong>We identified 15 observational (case-control and cross-sectional) studies for a total of 249,933 patients with psoriasis (49% with NAFLD) and 1,491,402 controls (36% with NAFLD). Psoriasis was associated with prevalent NAFLD (n = 11 studies; pooled random-effects odds ratio [OR] 1.96, 95% CI 1.70-2.26; I<sup>2</sup> = 97%, p < 0.01). Psoriatic patients with NAFLD had a higher mean psoriasis area and severity index (PASI) than their counterparts without NAFLD (n = 8 studies, pooled weighted mean difference: 3.93, 95% CI 2.01-5.84; I<sup>2</sup> = 88%, p < 0.01). The risk of NAFLD was marginally higher in patients with psoriatic arthritis than in those with psoriasis alone (n = 5 studies, pooled random-effects OR 1.83, 95% CI 0.98-3.43; I<sup>2</sup> = 64%, p = 0.03). Sensitivity analyses did not alter these findings. Funnel plot did not show any significant publication bias. A major limitation of the study was the high degree of heterogeneity across studies.</p><p><strong>Conclusion: </strong>Psoriasis is associated with prevalent NAFLD and this risk parallels the severity of psoriasis.</p>","PeriodicalId":15651,"journal":{"name":"Journal of Endocrinological Investigation","volume":"45 7","pages":"1277-1288"},"PeriodicalIF":5.4,"publicationDate":"2022-07-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9184411/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"39909574","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":2,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2022-06-17DOI: 10.1007/s40618-022-01833-3
A. Streb, P. Braga, R. F. D. DE MELO, L. J. Botelho, R. Maranhão, G. F. Del Duca
{"title":"Effects of combined physical exercise on plasma lipid variables, paraoxonase 1 activity, and inflammation parameters in adults with obesity: a randomized clinical trial","authors":"A. Streb, P. Braga, R. F. D. DE MELO, L. J. Botelho, R. Maranhão, G. F. Del Duca","doi":"10.1007/s40618-022-01833-3","DOIUrl":"https://doi.org/10.1007/s40618-022-01833-3","url":null,"abstract":"","PeriodicalId":15651,"journal":{"name":"Journal of Endocrinological Investigation","volume":"45 1","pages":"1991 - 1997"},"PeriodicalIF":5.4,"publicationDate":"2022-06-17","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"46044942","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":2,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2022-06-11DOI: 10.1007/s40618-022-01817-3
J. V. Gusmão-Silva, D. C. K. Lichtenecker, L. A. Ferreira, Í. Gois, R. Argeri, G. Gomes, M. Dias-da-Silva
{"title":"Body, metabolic and renal changes following cross-sex estrogen/progestogen therapy in a rodent model simulating its use by transwomen","authors":"J. V. Gusmão-Silva, D. C. K. Lichtenecker, L. A. Ferreira, Í. Gois, R. Argeri, G. Gomes, M. Dias-da-Silva","doi":"10.1007/s40618-022-01817-3","DOIUrl":"https://doi.org/10.1007/s40618-022-01817-3","url":null,"abstract":"","PeriodicalId":15651,"journal":{"name":"Journal of Endocrinological Investigation","volume":"45 1","pages":"1875 - 1885"},"PeriodicalIF":5.4,"publicationDate":"2022-06-11","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"48782877","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":2,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2022-06-10DOI: 10.1007/s40618-022-01826-2
A. De Leo, G. Vara, A. Paccapelo, C. Balacchi, V. Vicennati, L. Tucci, U. Pagotto, S. Selva, C. Ricci, L. Alberici, F. Minni, C. Nanni, F. Ambrosi, D. Santini, R. Golfieri, G. Di Dalmazi, C. Mosconi
{"title":"Computerized tomography texture analysis of pheochromocytoma: relationship with hormonal and histopathological data","authors":"A. De Leo, G. Vara, A. Paccapelo, C. Balacchi, V. Vicennati, L. Tucci, U. Pagotto, S. Selva, C. Ricci, L. Alberici, F. Minni, C. Nanni, F. Ambrosi, D. Santini, R. Golfieri, G. Di Dalmazi, C. Mosconi","doi":"10.1007/s40618-022-01826-2","DOIUrl":"https://doi.org/10.1007/s40618-022-01826-2","url":null,"abstract":"","PeriodicalId":15651,"journal":{"name":"Journal of Endocrinological Investigation","volume":"45 1","pages":"1935 - 1944"},"PeriodicalIF":5.4,"publicationDate":"2022-06-10","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"43834016","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":2,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2022-06-07DOI: 10.1007/s40618-022-01827-1
X. Ke, L. Duan, F. Gong, Y. Zhang, K. Deng, Y. Yao, L. Wang, F. Feng, B. Xing, H. Pan, H. Zhu
{"title":"A study on serum pro-neurotensin (PNT), furin, and zinc alpha-2-glycoprotein (ZAG) levels in patients with acromegaly","authors":"X. Ke, L. Duan, F. Gong, Y. Zhang, K. Deng, Y. Yao, L. Wang, F. Feng, B. Xing, H. Pan, H. Zhu","doi":"10.1007/s40618-022-01827-1","DOIUrl":"https://doi.org/10.1007/s40618-022-01827-1","url":null,"abstract":"","PeriodicalId":15651,"journal":{"name":"Journal of Endocrinological Investigation","volume":"45 1","pages":"1945 - 1954"},"PeriodicalIF":5.4,"publicationDate":"2022-06-07","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"47103529","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":2,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2022-06-01Epub Date: 2022-02-08DOI: 10.1007/s40618-022-01754-1
G Giuffrida, S Crisafulli, F Ferraù, A Fontana, Y Alessi, F Calapai, M Ragonese, N Luxi, S Cannavò, G Trifirò
Purpose: Cushing's disease (CD), 70% of endogenous hypercortisolism cases, is a rare disease caused by adrenocorticotropic hormone (ACTH)-secreting pituitary adenomas. To date, no systematic reviews and meta-analyses on its global epidemiology have been published. We provide a systematic review and meta-analysis of CD global epidemiology, also evaluating the quality of study reporting for the identified studies.
Methods: MEDLINE and EMBASE databases were searched for studies on CD epidemiology from inception until November 30th, 2020, including original observational studies in English about CD prevalence and/or incidence for well-defined geographic areas. Two reviewers independently extracted data and assessed reporting quality. CD prevalence/incidence pooled estimates were derived from a random-effects meta-analysis. Reporting quality was assessed using a STrengthening the Reporting of OBservational studies in Epidemiology (STROBE) checklist adapted for observational studies on rare diseases, heterogeneity using the Cochran's Q-test and its derived measure of inconsistency (I2).
Results: Thirteen studies were included. The pooled CD prevalence was 2.2 [95% CI 1.1-4.8] per 100,000, while the incidence rate was 0.24 [95% CI 0.15-0.33] per 100,000 person-years. For both parameters, considerable between-studies heterogeneity was found (I2 = 78.8% and 87.8%, respectively). The quality of study reporting was rated as medium for 11 (84.6%) studies and as low for 2 (15.4%).
Conclusion: Overall, our systematic meta-analysis demonstrated CD epidemiology to be similarly reported across different areas of the world, with some exceptions regarding regional differences or observation period intervals. Keeping into account the methodological differences between each paper, large-scale studies on CD epidemiology are warranted. Setting up national specific registries, based on standardized diagnostic and clinical parameters, with clearly defined selection and analysis criteria, and a strong cooperation between the scientific national societies for endocrinology is crucial to exclude other causes of variability (i.e. geographical differences due to other factors like (epi)genetic changes), and to support public health decision making.
目的:库欣病(Cushing's disease, CD)是一种由促肾上腺皮质激素(ACTH)分泌的垂体腺瘤引起的罕见疾病,占内源性高皮质醇症病例的70%。迄今为止,尚未发表关于其全球流行病学的系统综述和荟萃分析。我们对全球乳糜泻流行病学进行了系统回顾和荟萃分析,并对已确定的研究报告的质量进行了评估。方法:检索MEDLINE和EMBASE数据库从建立到2020年11月30日的CD流行病学研究,包括关于明确地理区域CD患病率和/或发病率的英文原始观察性研究。两名审稿人独立提取数据并评估报告质量。CD患病率/发病率汇总估计来自随机效应荟萃分析。采用适用于罕见病观察性研究的流行病学观察性研究报告强化检查表(STROBE)评估报告质量,采用Cochran’s q检验及其衍生的不一致性测量(I2)评估异质性。结果:纳入13项研究。合并CD患病率为每10万人2.2例[95% CI 1.1-4.8],发病率为每10万人年0.24例[95% CI 0.15-0.33]。对于这两个参数,研究间存在相当大的异质性(I2分别= 78.8%和87.8%)。11项研究报告的质量为中等(84.6%),2项研究报告的质量为低(15.4%)。结论:总体而言,我们的系统荟萃分析表明,除了区域差异或观察期间隔外,世界不同地区的CD流行病学报告相似。考虑到每篇论文的方法差异,对乳糜泻流行病学的大规模研究是有必要的。根据标准化的诊断和临床参数,建立具有明确定义的选择和分析标准的国家特定登记处,并在国家内分泌学科学学会之间进行强有力的合作,对于排除其他变异原因(即由于遗传变化等其他因素造成的地理差异)和支持公共卫生决策至关重要。
{"title":"Global Cushing's disease epidemiology: a systematic review and meta-analysis of observational studies.","authors":"G Giuffrida, S Crisafulli, F Ferraù, A Fontana, Y Alessi, F Calapai, M Ragonese, N Luxi, S Cannavò, G Trifirò","doi":"10.1007/s40618-022-01754-1","DOIUrl":"https://doi.org/10.1007/s40618-022-01754-1","url":null,"abstract":"<p><strong>Purpose: </strong>Cushing's disease (CD), 70% of endogenous hypercortisolism cases, is a rare disease caused by adrenocorticotropic hormone (ACTH)-secreting pituitary adenomas. To date, no systematic reviews and meta-analyses on its global epidemiology have been published. We provide a systematic review and meta-analysis of CD global epidemiology, also evaluating the quality of study reporting for the identified studies.</p><p><strong>Methods: </strong>MEDLINE and EMBASE databases were searched for studies on CD epidemiology from inception until November 30th, 2020, including original observational studies in English about CD prevalence and/or incidence for well-defined geographic areas. Two reviewers independently extracted data and assessed reporting quality. CD prevalence/incidence pooled estimates were derived from a random-effects meta-analysis. Reporting quality was assessed using a STrengthening the Reporting of OBservational studies in Epidemiology (STROBE) checklist adapted for observational studies on rare diseases, heterogeneity using the Cochran's Q-test and its derived measure of inconsistency (I<sup>2</sup>).</p><p><strong>Results: </strong>Thirteen studies were included. The pooled CD prevalence was 2.2 [95% CI 1.1-4.8] per 100,000, while the incidence rate was 0.24 [95% CI 0.15-0.33] per 100,000 person-years. For both parameters, considerable between-studies heterogeneity was found (I<sup>2</sup> = 78.8% and 87.8%, respectively). The quality of study reporting was rated as medium for 11 (84.6%) studies and as low for 2 (15.4%).</p><p><strong>Conclusion: </strong>Overall, our systematic meta-analysis demonstrated CD epidemiology to be similarly reported across different areas of the world, with some exceptions regarding regional differences or observation period intervals. Keeping into account the methodological differences between each paper, large-scale studies on CD epidemiology are warranted. Setting up national specific registries, based on standardized diagnostic and clinical parameters, with clearly defined selection and analysis criteria, and a strong cooperation between the scientific national societies for endocrinology is crucial to exclude other causes of variability (i.e. geographical differences due to other factors like (epi)genetic changes), and to support public health decision making.</p>","PeriodicalId":15651,"journal":{"name":"Journal of Endocrinological Investigation","volume":"45 6","pages":"1235-1246"},"PeriodicalIF":5.4,"publicationDate":"2022-06-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"39596408","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":2,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}