Marianna Beghini , Maximilian Pichler , Fiona Carolina Tinnefeld , Matthäus Metz , Dorothea Möslinger , Vassiliki Konstantopoulou , Johannes Spenger , Alexandra Kautzky-Willer , Florian Frommlet , Thomas Scherer , Miriam Hufgard-Leitner
{"title":"青春期依从性差是苯丙酮尿症患者失去随访的一个风险因素","authors":"Marianna Beghini , Maximilian Pichler , Fiona Carolina Tinnefeld , Matthäus Metz , Dorothea Möslinger , Vassiliki Konstantopoulou , Johannes Spenger , Alexandra Kautzky-Willer , Florian Frommlet , Thomas Scherer , Miriam Hufgard-Leitner","doi":"10.1016/j.ymgmr.2024.101087","DOIUrl":null,"url":null,"abstract":"<div><h3>Purpose</h3><p>A high rate of lost to follow-up (LTFU) in patients with phenylketonuria (PKU) represents a main challenge. In this study, we investigated potential risk factors for becoming LTFU related to adolescence as a critical period of life.</p></div><div><h3>Methods</h3><p>We retrospectively analyzed longitudinal data collected from 1993 to 2019 of patients diagnosed with classic PKU that were followed at our center during adolescence (14–18 y) and at least once in adulthood (>18 y). Patients who interrupted their contact with our center after the 18th birthday for at least 2 years were classified as LTFU. We performed a multivariate regression analysis to investigate following potential risk factors for becoming LTFU in adult life: sex, dietary compliance during adolescence assessed through the mean of the annual medians of phenylalanine plasma values, average number of contacts with the center during adolescence and age at first visit after the 18th birthday.</p></div><div><h3>Results</h3><p>93 patients (52 males, 41 females) were included in the study. 58% became LTFU during adulthood. The mean age at the last visit before becoming LTFU was 26.2 ± 5.1 years. In the multivariate Cox regression analysis we found that poor dietary compliance during adolescence was significantly associated with a higher risk of becoming LTFU during adulthood (<em>p</em>-value = 0.028).</p></div><div><h3>Discussion</h3><p>Adult patients who displayed poor treatment adherence during adolescence should be identified and carefully monitored to prevent loss of contact.</p></div>","PeriodicalId":18814,"journal":{"name":"Molecular Genetics and Metabolism Reports","volume":null,"pages":null},"PeriodicalIF":1.8000,"publicationDate":"2024-05-09","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.sciencedirect.com/science/article/pii/S2214426924000405/pdfft?md5=1508159e8abde38b8437ef90f76e7cfe&pid=1-s2.0-S2214426924000405-main.pdf","citationCount":"0","resultStr":"{\"title\":\"Poor adherence during adolescence is a risk factor for becoming lost-to-follow-up in patients with phenylketonuria\",\"authors\":\"Marianna Beghini , Maximilian Pichler , Fiona Carolina Tinnefeld , Matthäus Metz , Dorothea Möslinger , Vassiliki Konstantopoulou , Johannes Spenger , Alexandra Kautzky-Willer , Florian Frommlet , Thomas Scherer , Miriam Hufgard-Leitner\",\"doi\":\"10.1016/j.ymgmr.2024.101087\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<div><h3>Purpose</h3><p>A high rate of lost to follow-up (LTFU) in patients with phenylketonuria (PKU) represents a main challenge. In this study, we investigated potential risk factors for becoming LTFU related to adolescence as a critical period of life.</p></div><div><h3>Methods</h3><p>We retrospectively analyzed longitudinal data collected from 1993 to 2019 of patients diagnosed with classic PKU that were followed at our center during adolescence (14–18 y) and at least once in adulthood (>18 y). Patients who interrupted their contact with our center after the 18th birthday for at least 2 years were classified as LTFU. We performed a multivariate regression analysis to investigate following potential risk factors for becoming LTFU in adult life: sex, dietary compliance during adolescence assessed through the mean of the annual medians of phenylalanine plasma values, average number of contacts with the center during adolescence and age at first visit after the 18th birthday.</p></div><div><h3>Results</h3><p>93 patients (52 males, 41 females) were included in the study. 58% became LTFU during adulthood. The mean age at the last visit before becoming LTFU was 26.2 ± 5.1 years. In the multivariate Cox regression analysis we found that poor dietary compliance during adolescence was significantly associated with a higher risk of becoming LTFU during adulthood (<em>p</em>-value = 0.028).</p></div><div><h3>Discussion</h3><p>Adult patients who displayed poor treatment adherence during adolescence should be identified and carefully monitored to prevent loss of contact.</p></div>\",\"PeriodicalId\":18814,\"journal\":{\"name\":\"Molecular Genetics and Metabolism Reports\",\"volume\":null,\"pages\":null},\"PeriodicalIF\":1.8000,\"publicationDate\":\"2024-05-09\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"https://www.sciencedirect.com/science/article/pii/S2214426924000405/pdfft?md5=1508159e8abde38b8437ef90f76e7cfe&pid=1-s2.0-S2214426924000405-main.pdf\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Molecular Genetics and Metabolism Reports\",\"FirstCategoryId\":\"3\",\"ListUrlMain\":\"https://www.sciencedirect.com/science/article/pii/S2214426924000405\",\"RegionNum\":4,\"RegionCategory\":\"医学\",\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"\",\"PubModel\":\"\",\"JCR\":\"Q3\",\"JCRName\":\"GENETICS & HEREDITY\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Molecular Genetics and Metabolism Reports","FirstCategoryId":"3","ListUrlMain":"https://www.sciencedirect.com/science/article/pii/S2214426924000405","RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q3","JCRName":"GENETICS & HEREDITY","Score":null,"Total":0}
Poor adherence during adolescence is a risk factor for becoming lost-to-follow-up in patients with phenylketonuria
Purpose
A high rate of lost to follow-up (LTFU) in patients with phenylketonuria (PKU) represents a main challenge. In this study, we investigated potential risk factors for becoming LTFU related to adolescence as a critical period of life.
Methods
We retrospectively analyzed longitudinal data collected from 1993 to 2019 of patients diagnosed with classic PKU that were followed at our center during adolescence (14–18 y) and at least once in adulthood (>18 y). Patients who interrupted their contact with our center after the 18th birthday for at least 2 years were classified as LTFU. We performed a multivariate regression analysis to investigate following potential risk factors for becoming LTFU in adult life: sex, dietary compliance during adolescence assessed through the mean of the annual medians of phenylalanine plasma values, average number of contacts with the center during adolescence and age at first visit after the 18th birthday.
Results
93 patients (52 males, 41 females) were included in the study. 58% became LTFU during adulthood. The mean age at the last visit before becoming LTFU was 26.2 ± 5.1 years. In the multivariate Cox regression analysis we found that poor dietary compliance during adolescence was significantly associated with a higher risk of becoming LTFU during adulthood (p-value = 0.028).
Discussion
Adult patients who displayed poor treatment adherence during adolescence should be identified and carefully monitored to prevent loss of contact.
期刊介绍：
Molecular Genetics and Metabolism Reports is an open access journal that publishes molecular and metabolic reports describing investigations that use the tools of biochemistry and molecular biology for studies of normal and diseased states. In addition to original research articles, sequence reports, brief communication reports and letters to the editor are considered.
京公网安备 11010802042870号
京ICP备2023020795号-1
分享
求助内容：
应助结果提醒方式：
扫码关注我们
