Abstract：
OBJECTIVE: To evaluate the oral health-related quality of life (OHRQoL) of individuals diagnosed with Fanconi anemia (FA).
METHODS: A cross-sectional study was conducted with FA patients from two Brazilian referral centers. Participants underwent a complete dental, periodontal, and oral mucosa examination, as well as assessment of resting salivary flow. The short version of the Oral Health Impact Profile (OHIP-14) questionnaire was administered. Descriptive and bivariate analyses were performed, followed by multivariate analysis to examine the impact of independent variables on OHRQoL.
RESULTS: The study included 20 (57.1%) males and 15 (42.9%) females, with a mean age of 18.9 years. Oral leukoplakia (OL) was found in 18 individuals. The overall OHIP-14 score was 9.9 ± 10.5. Individuals aged ≥ 16 years had higher OHIP-14 scores, indicating worse OHRQoL for physical pain (p = 0.007), psychological discomfort (p = 0.001), physical disability (p = 0.03), psychological disability (p = 0.001), handicap (p = 0.004), and overall score (p = 0.007). Females reported more negative OHRQoL than males for physical pain (p = 0.02), psychological discomfort (p = 0.03), psychological disability (p = 0.009), and overall score (p = 0.02). Individuals with OL had an overall OHIP-14 score 1.83 times higher than those without OL (95% CI: 1.02-3.28; p = 0.04). Lower salivary flow correlated with higher overall OHIP-14 scores (95% CI: 0.14-0.84; p = 0.01).
CONCLUSIONS: This study represents the first attempt to evaluate OHRQoL in individuals with FA. The presence of OL and reduced salivary flow were identified as predictors of a negative impact on OHRQoL. It is imperative to integrate patients\' quality of life in the clinical treatment protocols for the FA population.
METHODS: A cross-sectional study was conducted with FA patients from two Brazilian referral centers. Participants underwent a complete dental, periodontal, and oral mucosa examination, as well as assessment of resting salivary flow. The short version of the Oral Health Impact Profile (OHIP-14) questionnaire was administered. Descriptive and bivariate analyses were performed, followed by multivariate analysis to examine the impact of independent variables on OHRQoL.
RESULTS: The study included 20 (57.1%) males and 15 (42.9%) females, with a mean age of 18.9 years. Oral leukoplakia (OL) was found in 18 individuals. The overall OHIP-14 score was 9.9 ± 10.5. Individuals aged ≥ 16 years had higher OHIP-14 scores, indicating worse OHRQoL for physical pain (p = 0.007), psychological discomfort (p = 0.001), physical disability (p = 0.03), psychological disability (p = 0.001), handicap (p = 0.004), and overall score (p = 0.007). Females reported more negative OHRQoL than males for physical pain (p = 0.02), psychological discomfort (p = 0.03), psychological disability (p = 0.009), and overall score (p = 0.02). Individuals with OL had an overall OHIP-14 score 1.83 times higher than those without OL (95% CI: 1.02-3.28; p = 0.04). Lower salivary flow correlated with higher overall OHIP-14 scores (95% CI: 0.14-0.84; p = 0.01).
CONCLUSIONS: This study represents the first attempt to evaluate OHRQoL in individuals with FA. The presence of OL and reduced salivary flow were identified as predictors of a negative impact on OHRQoL. It is imperative to integrate patients\' quality of life in the clinical treatment protocols for the FA population.
摘要：
目的：评估Fanconi贫血（FA）患者口腔健康相关生活质量（OHRQoL）。
方法：对来自两个巴西转诊中心的FA患者进行了一项横断面研究。参与者接受了完整的牙科检查，牙周,和口腔粘膜检查，以及静息唾液流量的评估。进行了简短版本的口腔健康影响概况（OHIP-14）问卷。进行了描述性和双变量分析，随后进行多变量分析，以检查独立变量对OHRQoL的影响。
结果：该研究包括20名（57.1％）男性和15名（42.9％）女性，平均年龄18.9岁。在18个人中发现了口腔白斑（OL）。OHIP-14总评分为9.9±10.5。年龄≥16岁的个体有更高的OHIP-14评分，表明身体疼痛的OHRQoL较差（p=0.007），心理不适（p=0.001），身体残疾(p=0.03)，心理障碍（p=0.001），障碍(p=0.004)，和总分（p=0.007）。女性在身体疼痛方面报告的OHRQoL阴性高于男性（p=0.02），心理不适（p=0.03），心理障碍（p=0.009），和总分（p=0.02）。具有OL的个体的总体OHIP-14得分比没有OL的个体高1.83倍(95%CI:1.02-3.28;p=0.04)。较低的唾液流量与较高的总OHIP-14评分相关(95%CI:0.14-0.84;p=0.01)。
结论：本研究首次尝试评估FA患者的OHRQoL。OL的存在和唾液流量减少被确定为对OHRQoL产生负面影响的预测因子。必须将患者的生活质量纳入FA人群的临床治疗方案。
方法：对来自两个巴西转诊中心的FA患者进行了一项横断面研究。参与者接受了完整的牙科检查，牙周,和口腔粘膜检查，以及静息唾液流量的评估。进行了简短版本的口腔健康影响概况（OHIP-14）问卷。进行了描述性和双变量分析，随后进行多变量分析，以检查独立变量对OHRQoL的影响。
结果：该研究包括20名（57.1％）男性和15名（42.9％）女性，平均年龄18.9岁。在18个人中发现了口腔白斑（OL）。OHIP-14总评分为9.9±10.5。年龄≥16岁的个体有更高的OHIP-14评分，表明身体疼痛的OHRQoL较差（p=0.007），心理不适（p=0.001），身体残疾(p=0.03)，心理障碍（p=0.001），障碍(p=0.004)，和总分（p=0.007）。女性在身体疼痛方面报告的OHRQoL阴性高于男性（p=0.02），心理不适（p=0.03），心理障碍（p=0.009），和总分（p=0.02）。具有OL的个体的总体OHIP-14得分比没有OL的个体高1.83倍(95%CI:1.02-3.28;p=0.04)。较低的唾液流量与较高的总OHIP-14评分相关(95%CI:0.14-0.84;p=0.01)。
结论：本研究首次尝试评估FA患者的OHRQoL。OL的存在和唾液流量减少被确定为对OHRQoL产生负面影响的预测因子。必须将患者的生活质量纳入FA人群的临床治疗方案。
