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Background <p>To retrospectively investigate the developmental outcomes at 3 years of age in patients with congenital diaphragmatic hernia (CDH) using a multicenter collaborative research approach.</p> Methods <p>We evaluated patients with CDH and no other malformations born between 2010 and 2016 in seven facilities in the Japanese CDH Research Group. The developmental quotient (DQ) at 3 years of age was evaluated using the Kyoto Scale of Psychological Development 2001, the most standardized scale in Japan. Factors associated with a DQ score &lt; 85 were also analyzed.</p> Results <p>Of 196 patients, developmental assessments at 3 years of age were performed in 132 patients (67%). Among these, 99 patients (75%) had a DQ score ≥ 85, 25 (19%) had a DQ score between 70 and 84, and 8 (6%) had a DQ score &lt; 70. Multivariate analysis showed that the observed/expected lung area-to-head circumference ratio was an independent predictor of a DQ score &lt; 85, with an adjusted odds ratio of 0.62 (95% confidence interval: 0.40–0.96; <i>p</i> = 0.03).</p> Conclusion <p>Generally, isolated CDH is associated with good developmental outcomes for survivors, even after intensive care. However, there is a risk of neurodevelopmental impairment if pulmonary hypoplasia is present.</p> Impact <p><UnorderedList Mark="Bullet"> <ItemContent> <p>This research highlights the observed/expected lung area-to-head circumference ratio (o/e LHR) as a crucial indicator to predict neurodevelopmental outcomes in 3-year-old children diagnosed with isolated congenital diaphragmatic hernia (CDH).</p> </ItemContent> <ItemContent> <p>Our results provide robust evidence from a large multicenter cohort, emphasizing the importance of o/e LHR in early risk stratification and prolonged neurodevelopmental follow-up.</p> </ItemContent> <ItemContent> <p>These findings highlight the need for comprehensive management and tailored follow-up care in CDH patients, potentially improving clinical protocols and enhancing the quality of life and outcomes for affected children.</p> </ItemContent> </UnorderedList></p>

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Isolated congenital diaphragmatic hernia and three-year neurodevelopmental outcomes

  • 期刊论文

  • 出版模式：

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  • 发表日期：

    2025年1月23日

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  • Katsuaki Toyoshima,
  • Hirosato Aoki,
  • Kaoru Katsumata,
  • Yoshiaki Sato,
  • Hirosuke Inoue,
  • Miharu Ito,
  • Shoichiro Amari,
  • Hidehiko Maruyama,
  • Hitomi Arahori,
  • Takuya Kondo,
  • Kiyokazu Kim,
  • Masaya Yamoto,
  • Tomoko Saito,
  • Hiroomi Okuyama,
  • Noriaki Usui,
  • Keita Terui,
  • Kouji Nagata

摘要

Background

To retrospectively investigate the developmental outcomes at 3 years of age in patients with congenital diaphragmatic hernia (CDH) using a multicenter collaborative research approach.

Methods

We evaluated patients with CDH and no other malformations born between 2010 and 2016 in seven facilities in the Japanese CDH Research Group. The developmental quotient (DQ) at 3 years of age was evaluated using the Kyoto Scale of Psychological Development 2001, the most standardized scale in Japan. Factors associated with a DQ score < 85 were also analyzed.

Results

Of 196 patients, developmental assessments at 3 years of age were performed in 132 patients (67%). Among these, 99 patients (75%) had a DQ score ≥ 85, 25 (19%) had a DQ score between 70 and 84, and 8 (6%) had a DQ score < 70. Multivariate analysis showed that the observed/expected lung area-to-head circumference ratio was an independent predictor of a DQ score < 85, with an adjusted odds ratio of 0.62 (95% confidence interval: 0.40–0.96; p = 0.03).

Conclusion

Generally, isolated CDH is associated with good developmental outcomes for survivors, even after intensive care. However, there is a risk of neurodevelopmental impairment if pulmonary hypoplasia is present.

Impact

This research highlights the observed/expected lung area-to-head circumference ratio (o/e LHR) as a crucial indicator to predict neurodevelopmental outcomes in 3-year-old children diagnosed with isolated congenital diaphragmatic hernia (CDH).

Our results provide robust evidence from a large multicenter cohort, emphasizing the importance of o/e LHR in early risk stratification and prolonged neurodevelopmental follow-up.

These findings highlight the need for comprehensive management and tailored follow-up care in CDH patients, potentially improving clinical protocols and enhancing the quality of life and outcomes for affected children.