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Endoscopic Versus Open Repair for Craniosynostosis in Infants Using Propensity Score Matching to Compare Outcomes: A Multicenter Study from the Pediatric Craniofacial Collaborative Group

Thompson, Douglas R. MD*; Zurakowski, David MS, PhD; Haberkern, Charles M. MD, MPH*‡; Stricker, Paul A. MD§; Meier, Petra M. MD, DEAA; Bannister, Carolyn; Benzon, Hubert; Binstock, Wendy; Bosenberg, Adrian; Brzenski, Alyssa; Budac, Stefan; Busso, Veronica; Capehart, Samantha; Chiao, Franklin; Cladis, Franklyn; Collins, Michael; Cusick, Jordan; Dabek, Rachel; Dalesio, Nicholas; Falcon, Ricardo; Fernandez, Allison; Fernandez, Patrick; Fiadjoe, John; Gangadharan, Meera; Gentry, Katherine; Glover, Chris; Goobie, Susan; Gries, Heike; Griffin, Allison; Groenewald, Cornelius Botha; Hajduk, John; Hall, Rebecca; Hansen, Jennifer; Hetmaniuk, Mali; Hsieh, Vincent; Huang, Henry; Ingelmo, Pablo; Ivanova, Iskra; Jain, Ranu; Koh, Jeffrey; Kowalczyk-Derderian, Courtney; Kugler, Jane; Labovsky, Kristen; Martinez, José Luis; Mujallid, Razaz; Muldowney, Bridget; Nguyen, Kim-Phuong; Nguyen, Thanh; Olutuye, Olutoyin; Soneru, Codruta; Petersen, Timothy; Poteet-Schwartz, Kim; Reddy, Srijaya; Reid, Russell; Ricketts, Karene; Rubens, Daniel; Skitt, Rochelle; Sohn, Lisa; Staudt, Susan; Sung, Wai; Syed, Tariq; Szmuk, Peter; Taicher, Brad; Tetreault, Lisa; Watts, Rheana; Wong, Karen; Young, Vanessa; Zamora, LillianThe Pediatric Craniofacial Collaborative Group

doi: 10.1213/ANE.0000000000002454
Neuroscience and Neuroanesthesiology: Original Clinical Research Report

BACKGROUND: The North American Pediatric Craniofacial Collaborative Group (PCCG) established the Pediatric Craniofacial Surgery Perioperative Registry to evaluate outcomes in infants and children undergoing craniosynostosis repair. The goal of this multicenter study was to utilize this registry to assess differences in blood utilization, intensive care unit (ICU) utilization, duration of hospitalization, and perioperative complications between endoscopic-assisted (ESC) and open repair in infants with craniosynostosis. We hypothesized that advantages of ESC from single-center studies would be validated based on combined data from a large multicenter registry.

METHODS: Thirty-one institutions contributed data from June 2012 to September 2015. We analyzed 1382 infants younger than 12 months undergoing open (anterior and/or posterior cranial vault reconstruction, modified-Pi procedure, or strip craniectomy) or endoscopic craniectomy. The primary outcomes included transfusion data, ICU utilization, hospital length of stay, and perioperative complications; secondary outcomes included anesthesia and surgical duration. Comparison of unmatched groups (ESC: N = 311, open repair: N = 1071) and propensity score 2:1 matched groups (ESC: N = 311, open repair: N = 622) were performed by conditional logistic regression analysis.

RESULTS: Imbalances in baseline age and weight are inherent due to surgical selection criteria for ESC. Quality of propensity score matching in balancing age and weight between ESC and open groups was assessed by quintiles of the propensity scores. Analysis of matched groups confirmed significantly reduced utilization of blood (26% vs 81%, P < .001) and coagulation (3% vs 16%, P < .001) products in the ESC group compared to the open group. Median blood donor exposure (0 vs 1), anesthesia (168 vs 248 minutes) and surgical duration (70 vs 130 minutes), days in ICU (0 vs 2), and hospital length of stay (2 vs 4) were all significantly lower in the ESC group (all P < .001). Median volume of red blood cell administered was significantly lower in ESC (19.6 vs 26.9 mL/kg, P = .035), with a difference of approximately 7 mL/kg less for the ESC (95% confidence interval for the difference, 3–12 mL/kg), whereas the median volume of coagulation products was not significantly different between the 2 groups (21.2 vs 24.6 mL/kg, P = .73). Incidence of complications including hypotension requiring treatment with vasoactive agents (3% vs 4%), venous air embolism (1%), and hypothermia, defined as <35°C (22% vs 26%), was similar between the 2 groups, whereas postoperative intubation was significantly higher in the open group (2% vs 10%, P < .001).

CONCLUSIONS: This multicenter study of ESC versus open craniosynostosis repair represents the largest comparison to date. It demonstrates striking advantages of ESC for young infants that may result in improved clinical outcomes, as well as increased safety.

From the *Department of Anesthesiology and Pain Medicine, University of Washington-Seattle Children’s Hospital, Seattle, Washington; Department of Anesthesiology, Perioperative and Pain Medicine, Boston Children’s Hospital, Harvard Medical School, Boston, Massachusetts; Department of Pediatrics (adj.), University of Washington-Seattle Children’s Hospital, Seattle, Washington; and §Department of Anesthesiology and Critical Care, The Children’s Hospital of Philadelphia and the Perelman School of Medicine at the University of Pennsylvania, Philadelphia, Pennsylvania.

cbannis@emory.edu

HBenzon@luriechildrens.org

WBinstock@dacc.uchicago.edu

adrian.bosenberg@seattlechildrens.org

abrzenski@ucsd.edu

Stefan.budac@seattlechildrens.org

Veronica.Busso@cchmc.org

sjcapeha@texaschildrens.org

fchiao@gmail.com

cladfp@upmc.edu

Michael.collins@seattlechildrens.org

cusick@ohsu.edu

Rachel.Dabek@childrens.harvard.edu

ndalesi1@jhmi.edu

rfalcon@salud.unm.edu

aferna27@jhmi.edu

Patrick.Fernandez@childrenscolorado.org

fiadjoej@email.chop.edu

meeraganga@gmail.com

Katherine.gentry@seattlechildrens.org

cdglover@texaschildrens.org

Susan.Goobie@childrens.harvard.edu

griesh@ohsu.edu

Allison.Griffin@UTSouthwestern.edu

cornelius.groenewald@seattlechildrens.org

JHajduk@luriechildrens.org

hallreb@ohsu.edu

jhansen4@kumc.edu

Mali.Hetmaniuk@seattlechildrens.org

vincent.hsieh@seattlechildrens.org

hxhuang@texaschildrens.org

Pablo.Ingelmo@MUHC.MCGILL.CA

Iskra.ivanova@seattlechildrens.org

Ranu.Jain@uth.tmc.edu

kohj@ohsu.edu

Courtney.Derderian@childrens.com

jakugler@childrensomaha.org

klabovsky@mcw.edu

jose.luis.martinez.hsj@ssssgouv.qc.ca

rhjm2004@yahoo.com

blmuldowney@wisc.edu

ktn@bcm.tmc.edu

thanh.t.nguyen.1@vanderbilt.edu

oaolutoy@texaschildrens.org

csoneru@salud.unm.edu

timpetersen@salud.unm.edu

KLPoteetschwartz@uams.edu

SReddy@childrensnational.org

rreid@surgery.bsd.uchicago.edu

kricketts@aims.unc.edu

daniel.rubens@seattlechildrens.org

Rochelle.Skitt@childrenscolorado.org

sohnlis@yahoo.com

sstaudt@mcw.edu

sungw@ccf.org

Tariq.A.Syed@uth.tmc.edu

pszmuk@gmail.com

brad.taicher@duke.edu

ltetrea2@jhmi.edu

rheana.watts@seattlechildrens.org

karen.wong@seattlechildrens.org

Vanessa.Young@childrens.harvard.edu

lzamora1@jhmi.edu

Published ahead of print September 14, 2017.

Accepted for publication July 25, 2017.

∥ Collaborators’ names and institutions are listed in Appendices 1 and 2.

Funding: This work was solely supported by the Department of Anesthesiology and Critical Care, The Children’s Hospital of Philadelphia, and the Department of Anesthesiology, Perioperative and Pain Medicine, Boston Children’s Hospital.

The authors declare no conflicts of interest.

Supplemental digital content is available for this article. Direct URL citations appear in the printed text and are provided in the HTML and PDF versions of this article on the journal’s website.

Reprints will not be available from the authors.

Address correspondence to Petra M. Meier, MD, DEAA, Department of Anesthesiology, Perioperative and Pain Medicine, Boston Children’s Hospital, Harvard Medical School, 300 Longwood Ave, Boston, MA 02115. Address e-mail to petra.meier-haran@childrens.harvard.edu.

© 2018 International Anesthesia Research Society
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