Beghini Marianna, Pichler Maximilian, Tinnefeld Fiona Carolina, Metz Matthäus, Möslinger Dorothea, Konstantopoulou Vassiliki, Spenger Johannes, Kautzky-Willer Alexandra, Frommlet Florian, Scherer Thomas, Hufgard-Leitner Miriam
Division of Endocrinology and Metabolism, Department of Internal Medicine III, Medical University of Vienna, Währinger Gürtel 18-20, 1090 Vienna, Austria.
Department of Paediatrics and Adolescent Medicine, Medical University of Vienna, Währinger Gürtel 18-20, 1090 Vienna, Austria.
Mol Genet Metab Rep. 2024 May 9;39:101087. doi: 10.1016/j.ymgmr.2024.101087. eCollection 2024 Jun.
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.
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.
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 (-value = 0.028).
Adult patients who displayed poor treatment adherence during adolescence should be identified and carefully monitored to prevent loss of contact.
苯丙酮尿症（PKU）患者中高失访率是一项主要挑战。在本研究中，我们调查了与青春期这一关键生命阶段相关的失访潜在风险因素。
我们回顾性分析了1993年至2019年收集的纵向数据，这些数据来自于在我们中心接受随访的青春期（14 - 18岁）及成年期（>18岁）至少随访一次的经典PKU确诊患者。18岁生日后与我们中心中断联系至少2年的患者被归类为失访。我们进行了多变量回归分析，以研究成年期失访的以下潜在风险因素：性别、通过苯丙氨酸血浆值年度中位数均值评估的青春期饮食依从性、青春期与中心的平均接触次数以及18岁生日后的首次就诊年龄。
93例患者（52例男性，41例女性）纳入研究。58%在成年期失访。失访前最后一次就诊的平均年龄为26.2±5.1岁。在多变量Cox回归分析中，我们发现青春期饮食依从性差与成年期失访风险较高显著相关（P值 = 0.028）。
应识别出青春期治疗依从性差的成年患者并对其进行仔细监测，以防止失去联系。
Zotero 插件