Neurodevelopmental Outcomes in Children After Fetoscopic Endoluminal Tracheal Occlusion for Severe Congenital Diaphragmatic Hernia: Results From a Multidisciplinary Clinic

被引:2
作者
Sferra, Shelby R. [1 ]
Penikis, Annalise B. [1 ]
Guo, Matthew [1 ]
Baschat, Ahmet A. [2 ]
Mogayzel, Peter J. [3 ]
Burton, Vera Joanna [4 ]
Kunisaki, Shaun M. [1 ,5 ]
机构
[1] Johns Hopkins Univ, Sch Med, Dept Surg, Div Gen Pediat Surg, 1800 Orleans St, Baltimore, MD 21287 USA
[2] Johns Hopkins Univ, Johns Hopkins Ctr Fetal Therapy, Sch Med, Dept Gynecol & Obstet, 600 N Wolfe St, Baltimore, MD 21287 USA
[3] Johns Hopkins Univ, Sch Med, Dept Pediat, Eudowood Div Pediat Pulm, 200 N Wolfe St, Baltimore, MD 21287 USA
[4] Johns Hopkins Univ, Sch Med, Dept Neurol, Neurol & Dev Med, 801 N Broadway, Baltimore, MD 21205 USA
[5] Johns Hopkins Univ, Sch Med, Div Gen Pediat Surg, 1800 Orleans St,Suite 7353, Baltimore, MD 21287 USA
关键词
Congenital diaphragmatic hernia; Fetal tracheal occlusion; Fetal therapy; Outcomes; EXTRACORPOREAL MEMBRANE-OXYGENATION; SURGICAL OUTCOMES; FOLLOW-UP; SURVIVORS; INFANTS;
D O I
10.1016/j.jpedsurg.2024.03.041
中图分类号
R72 [儿科学];
学科分类号
100202 ;
摘要
Background: We compared early neurodevelopmental morbidity in young children with severe CDH who underwent FETO to those without fetal therapy. Methods: We conducted a prospective study of severe CDH patients undergoing FETO (n = 18) at a single North American center from 2015 to 2021 (NCT02710968). Outpatient survivors (n = 12) were evaluated by a multidisciplinary team and compared to expectantly managed CDH patients. Neurodevelopmental outcomes were assessed using the Capute Scales [Clinical Linguistic and Auditory Milestone Scales (CLAMS) and Cognitive Adaptive Test (CAT)], with a developmental quotient (DQ) < 85 indicative of atrisk for delay. Results: At one year, 58% (n = 7) of FETO patients underwent evaluation, with notable concern for language delay (CLAMS median DQ, 80.1 [interquartile range, 67.6-86.7]). FETO scores improved by 24months, whereas high severity/non-FETO scores declined [CLAMS median DQ (Difference in DQ), 92.3 (+12.2) vs. 77.1 (-13.4), respectively; p = 0.049]. On the initial CAT, FETO patients had concern for visual motor and problem-solving delays, with a median DQ of 81.3 (62.1-89.4). At 24-months, FETO patients had improving scores [Median CAT DQ, 90.8 (+9.5)], whereas high severity/non-FETO [87.5 (-3.0), p = 0.28] had declining scores. Conclusion: These initial data suggest that FETO is associated with favorable neurodevelopmental outcomes at 24-months compared to severe CDH under expectant management. Level of Evidence: III.
引用
收藏
页码:1271 / 1276
页数:6
相关论文
共 36 条
  • [1] Single-Center Outcome of Fetoscopic Tracheal Balloon Occlusion for Severe Congenital Diaphragmatic Hernia
    Baschat, Ahmet A.
    Rosner, Mara
    Millard, Sarah E.
    Murphy, Jamie D.
    Blakemore, Karin J.
    Keiser, Amaris M.
    Kearney, Jennifer
    Bullard, Janine
    Nogee, Lawrence M.
    Bembea, Melania
    Jelin, Eric B.
    Miller, Jena L.
    [J]. OBSTETRICS AND GYNECOLOGY, 2020, 135 (03) : 511 - 521
  • [2] Neurodevelopmental outcome in congenital diaphragmatic hernia survivors: role of ventilatory time
    Bevilacqua, Francesca
    Morini, Francesco
    Zaccara, Antonio
    Valfre, Laura
    Capolupo, Irma
    Bagolan, Pietro
    Aite, Lucia
    [J]. JOURNAL OF PEDIATRIC SURGERY, 2015, 50 (03) : 394 - 398
  • [3] Neurodevelopmental outcomes in CDH survivors: A single institution's experience
    Church, Joseph T.
    Mon, Rodrigo
    Wright, Tiffany
    Coughlin, Megan A.
    Ladino-Torres, Maria
    Tapley, Christopher
    Bowen, Heather
    Matusko, Niki
    Mychaliska, George B.
    [J]. JOURNAL OF PEDIATRIC SURGERY, 2018, 53 (06) : 1087 - 1091
  • [4] Survival of severe congenital diaphragmatic hernia has morbid consequences
    Cortes, RA
    Keller, RL
    Townsend, T
    Harrison, MR
    Farmer, DL
    Lee, HM
    Piecuch, RE
    Leonard, CH
    Hetherton, M
    Bisgaard, R
    Nobuhara, KK
    [J]. JOURNAL OF PEDIATRIC SURGERY, 2005, 40 (01) : 36 - 45
  • [5] Rate and Risk Factors Associated with Autism Spectrum Disorder in Congenital Diaphragmatic Hernia
    Danzer, Enrico
    Hoffman, Casey
    D'Agostino, Jo Ann
    Miller, Judith S.
    Waqar, Lindsay N.
    Gerdes, Marsha
    Bernbaum, Judy C.
    Rosenthal, Hannah
    Rintoul, Natalie E.
    Herkert, Lisa M.
    Peranteau, William H.
    Flake, Alan W.
    Adzick, N. Scott
    Hedrick, Holly L.
    [J]. JOURNAL OF AUTISM AND DEVELOPMENTAL DISORDERS, 2018, 48 (06) : 2112 - 2121
  • [6] Short-Term Neurodevelopmental Outcome in Congenital Diaphragmatic Hernia: The Impact of Extracorporeal Membrane Oxygenation and Timing of Repair
    Danzer, Enrico
    Hoffman, Casey
    D'Agostino, Jo Ann
    Connelly, James T.
    Waqar, Lindsay N.
    Gerdes, Marsha
    Bernbaum, Judy
    Rintoul, Natalie E.
    Herkert, Lisa M.
    Peranteau, William H.
    Flake, Alan W.
    Adzick, N. Scott
    Hedrick, Holly L.
    [J]. PEDIATRIC CRITICAL CARE MEDICINE, 2018, 19 (01) : 64 - 74
  • [7] Neurodevelopmental outcomes at 5 years of age in congenital diaphragmatic hernia
    Danzer, Enrico
    Hoffman, Casey
    D'Agostino, Jo Ann
    Gerdes, Marsha
    Bernbaum, Judy
    Antiel, Ryan M.
    Rintoul, Natalie E.
    Herkert, Lisa M.
    Flake, Alan W.
    Adzick, N. Scott
    Hedrick, Holly L.
    [J]. JOURNAL OF PEDIATRIC SURGERY, 2017, 52 (03) : 437 - 443
  • [8] Danzer Enrico, 2014, World J Clin Pediatr, V3, P30, DOI 10.5409/wjcp.v3.i3.30
  • [9] Preschool neurological assessment in congenital diaphragmatic hernia survivors: Outcome and perinatal factors associated with neurodevelopmental impairment
    Danzer, Enrico
    Gerdes, Marsha
    D'Agostino, Jo Ann
    Partridge, Emily A.
    Hoffman-Craven, Casey H.
    Bernbaum, Judy
    Rintoul, Natalie E.
    Flake, Alan W.
    Adzick, N. Scott
    Hedrick, Holly L.
    [J]. EARLY HUMAN DEVELOPMENT, 2013, 89 (06) : 393 - 400
  • [10] Abnormal brain development and maturation on magnetic resonance imaging in survivors of severe congenital diaphragmatic hernia
    Danzer, Enrico
    Zarnow, Deborah
    Gerdes, Marsha
    D'Agostino, Jo Ann
    Siegle, Jennifer
    Bebbington, Michael W.
    Flake, Alan W.
    Adzick, N. Scott
    Hedrick, Holly L.
    [J]. JOURNAL OF PEDIATRIC SURGERY, 2012, 47 (03) : 453 - 461