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Pediatric neurology · 2025

Navigating the Emotional and Practical Challenges of Newborn Screening for Late-Onset Pompe Disease: Insights From Parental Perspectives.

СтатьяОбзорНазвание и аннотация: Перевод в очереди
Обработка статьи · этапы, ошибки и управление
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Boueri Myriam, Paltzer Allison, Huggins Erin, Linebaugh Ellen, Zvejnieks Debera, Doxey Jessica, Spiridigliozzi Gail, Jung Seung-Hye, Kishnani Priya S

Аннотация

Pompe disease (PD), an autosomal recessive lysosomal disorder, results in glycogen accumulation in muscle cells, leading to progressive muscle weakness and respiratory insufficiency. Newborn screening (NBS) has improved outcomes for infantile-onset PD by enabling early diagnosis and intervention with enzyme replacement therapy. NBS also identifies late-onset PD (LOPD) cases, wherein children have a wide clinical spectrum and may remain asymptomatic for years, placing families in uncertainty as "patients-in-waiting." This study explores parental experiences following an LOPD diagnosis through NBS to identify gaps in support systems and improve care delivery. Parents of 42 children diagnosed with LOPD through NBS completed a survey regarding their diagnostic experiences, care access, anxiety, and health care professionals' (HCPs') roles. Survey responses were analyzed using descriptive statistics, thematic analysis, and Kruskal-Wallis tests. Parents valued clear guidance and condition-specific information when receiving NBS results. However, many reported insufficient support and HCP's limited LOPD knowledge. About 70.7% experienced reduced anxiety following the LOPD diagnosis, attributed to increased knowledge, supportive health care teams, and their child's stable health, although uncertainty persisted. Among those who saw an HCP, 71.9% reported positive impacts, including improved understanding and mental health support, although 19% thought counseling or information provided lacked clarity or actionable resources. Timely communication with knowledgeable HCPs and multidisciplinary support can potentially reduce the psychosocial burden on families receiving positive NBS results. Efforts should prioritize creating more resources for HCPs and improving communication to ensure consistent compassionate care.

Предметные рубрики MeSH

ИП Урсолов Владлен Сергеевич

ИНН 500118482352 · ОГРНИП 314500117700049

Реквизиты и контакты