Shachi Srivatsa, Greg Metzger, Kyle Horvath, Kelli Patterson, Megan Scruggs, Tran Bourgeois, Joseph Stanek, Peter C. Minneci, Jennifer H. Aldrink, Keri A. Streby
{"title":"高危神经母细胞瘤患者肾上腺功能不全的发生率：单机构分析。","authors":"Shachi Srivatsa, Greg Metzger, Kyle Horvath, Kelli Patterson, Megan Scruggs, Tran Bourgeois, Joseph Stanek, Peter C. Minneci, Jennifer H. Aldrink, Keri A. Streby","doi":"10.1002/pbc.31543","DOIUrl":null,"url":null,"abstract":"<div>\n \n \n <section>\n \n <h3> Background</h3>\n \n <p>Neuroblastoma is the most common extracranial solid tumor in children, with about half of cases classified as high risk. Treatment varies by risk level, with high-risk patients undergoing aggressive multimodal therapy. While long-term survival has improved, survivors face significant risks of late treatment effects, including adrenal insufficiency. This study investigates the incidence of adrenal insufficiency among neuroblastoma patients of varying disease risk.</p>\n </section>\n \n <section>\n \n <h3> Procedure</h3>\n \n <p>This retrospective cohort study at a single tertiary children's hospital reviewed records from 1998 to 2021, identifying 370 neuroblastoma patients, with 137 having complete risk stratification. The primary outcome was the incidence of adrenal insufficiency, diagnosed via clinical evaluation, and compared by risk group.</p>\n </section>\n \n <section>\n \n <h3> Results</h3>\n \n <p>Among 137 patients with neuroblastoma, nine (12.0%) high-risk and three (4.9%) non-high-risk patients were diagnosed with adrenal insufficiency. The cumulative incidence of adrenal insufficiency at 3 years after neuroblastoma diagnosis was 9.9% in high-risk and 3.5% in non-high-risk patients. Among high-risk neuroblastoma patients with adrenal insufficiency, median time to adrenal insufficiency diagnosis was 10.2 months from neuroblastoma diagnosis, with cases more commonly occurring in patients with adrenal primary tumors (<i>p</i> = 0.0234). There were no other significant differences in demographic or clinical characteristics between high-risk patients with and without adrenal insufficiency.</p>\n </section>\n \n <section>\n \n <h3> Conclusions</h3>\n \n <p>Adrenal insufficiency occurs more commonly in children with high-risk neuroblastoma than in non-high-risk cases, particularly those with adrenal primary tumors. These findings underscore the need for vigilant monitoring and screening for adrenal insufficiency during and after treatment. Future research should include larger, multi-institutional cohorts to better understand risk factors and optimize screening protocols.</p>\n </section>\n </div>","PeriodicalId":19822,"journal":{"name":"Pediatric Blood & Cancer","volume":"72 4","pages":""},"PeriodicalIF":2.4000,"publicationDate":"2025-01-21","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://onlinelibrary.wiley.com/doi/epdf/10.1002/pbc.31543","citationCount":"0","resultStr":"{\"title\":\"Incidence of Adrenal Insufficiency in Patients With High-Risk Neuroblastoma: A Single-Institution Analysis\",\"authors\":\"Shachi Srivatsa, Greg Metzger, Kyle Horvath, Kelli Patterson, Megan Scruggs, Tran Bourgeois, Joseph Stanek, Peter C. Minneci, Jennifer H. Aldrink, Keri A. Streby\",\"doi\":\"10.1002/pbc.31543\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<div>\\n \\n \\n <section>\\n \\n <h3> Background</h3>\\n \\n <p>Neuroblastoma is the most common extracranial solid tumor in children, with about half of cases classified as high risk. Treatment varies by risk level, with high-risk patients undergoing aggressive multimodal therapy. While long-term survival has improved, survivors face significant risks of late treatment effects, including adrenal insufficiency. This study investigates the incidence of adrenal insufficiency among neuroblastoma patients of varying disease risk.</p>\\n </section>\\n \\n <section>\\n \\n <h3> Procedure</h3>\\n \\n <p>This retrospective cohort study at a single tertiary children's hospital reviewed records from 1998 to 2021, identifying 370 neuroblastoma patients, with 137 having complete risk stratification. The primary outcome was the incidence of adrenal insufficiency, diagnosed via clinical evaluation, and compared by risk group.</p>\\n </section>\\n \\n <section>\\n \\n <h3> Results</h3>\\n \\n <p>Among 137 patients with neuroblastoma, nine (12.0%) high-risk and three (4.9%) non-high-risk patients were diagnosed with adrenal insufficiency. The cumulative incidence of adrenal insufficiency at 3 years after neuroblastoma diagnosis was 9.9% in high-risk and 3.5% in non-high-risk patients. Among high-risk neuroblastoma patients with adrenal insufficiency, median time to adrenal insufficiency diagnosis was 10.2 months from neuroblastoma diagnosis, with cases more commonly occurring in patients with adrenal primary tumors (<i>p</i> = 0.0234). There were no other significant differences in demographic or clinical characteristics between high-risk patients with and without adrenal insufficiency.</p>\\n </section>\\n \\n <section>\\n \\n <h3> Conclusions</h3>\\n \\n <p>Adrenal insufficiency occurs more commonly in children with high-risk neuroblastoma than in non-high-risk cases, particularly those with adrenal primary tumors. These findings underscore the need for vigilant monitoring and screening for adrenal insufficiency during and after treatment. Future research should include larger, multi-institutional cohorts to better understand risk factors and optimize screening protocols.</p>\\n </section>\\n </div>\",\"PeriodicalId\":19822,\"journal\":{\"name\":\"Pediatric Blood & Cancer\",\"volume\":\"72 4\",\"pages\":\"\"},\"PeriodicalIF\":2.4000,\"publicationDate\":\"2025-01-21\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"https://onlinelibrary.wiley.com/doi/epdf/10.1002/pbc.31543\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Pediatric Blood & Cancer\",\"FirstCategoryId\":\"3\",\"ListUrlMain\":\"https://onlinelibrary.wiley.com/doi/10.1002/pbc.31543\",\"RegionNum\":3,\"RegionCategory\":\"医学\",\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"\",\"PubModel\":\"\",\"JCR\":\"Q2\",\"JCRName\":\"HEMATOLOGY\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Pediatric Blood & Cancer","FirstCategoryId":"3","ListUrlMain":"https://onlinelibrary.wiley.com/doi/10.1002/pbc.31543","RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q2","JCRName":"HEMATOLOGY","Score":null,"Total":0}
Incidence of Adrenal Insufficiency in Patients With High-Risk Neuroblastoma: A Single-Institution Analysis
Background
Neuroblastoma is the most common extracranial solid tumor in children, with about half of cases classified as high risk. Treatment varies by risk level, with high-risk patients undergoing aggressive multimodal therapy. While long-term survival has improved, survivors face significant risks of late treatment effects, including adrenal insufficiency. This study investigates the incidence of adrenal insufficiency among neuroblastoma patients of varying disease risk.
Procedure
This retrospective cohort study at a single tertiary children's hospital reviewed records from 1998 to 2021, identifying 370 neuroblastoma patients, with 137 having complete risk stratification. The primary outcome was the incidence of adrenal insufficiency, diagnosed via clinical evaluation, and compared by risk group.
Results
Among 137 patients with neuroblastoma, nine (12.0%) high-risk and three (4.9%) non-high-risk patients were diagnosed with adrenal insufficiency. The cumulative incidence of adrenal insufficiency at 3 years after neuroblastoma diagnosis was 9.9% in high-risk and 3.5% in non-high-risk patients. Among high-risk neuroblastoma patients with adrenal insufficiency, median time to adrenal insufficiency diagnosis was 10.2 months from neuroblastoma diagnosis, with cases more commonly occurring in patients with adrenal primary tumors (p = 0.0234). There were no other significant differences in demographic or clinical characteristics between high-risk patients with and without adrenal insufficiency.
Conclusions
Adrenal insufficiency occurs more commonly in children with high-risk neuroblastoma than in non-high-risk cases, particularly those with adrenal primary tumors. These findings underscore the need for vigilant monitoring and screening for adrenal insufficiency during and after treatment. Future research should include larger, multi-institutional cohorts to better understand risk factors and optimize screening protocols.
期刊介绍：
Pediatric Blood & Cancer publishes the highest quality manuscripts describing basic and clinical investigations of blood disorders and malignant diseases of childhood including diagnosis, treatment, epidemiology, etiology, biology, and molecular and clinical genetics of these diseases as they affect children, adolescents, and young adults. Pediatric Blood & Cancer will also include studies on such treatment options as hematopoietic stem cell transplantation, immunology, and gene therapy.
求助内容：
应助结果提醒方式：
京公网安备 11010802042870号
