2015 HSC Section 1 Book of Articles

Reprinted by permission of Otolaryngol Head Neck Surg. 2014; 151(5):811-818.

Original Research—Health Policy and Economics

Otolaryngology– Head and Neck Surgery 2014, Vol. 151(5) 811–818 American Academy of Otolaryngology—Head and Neck

Cost Analysis of Mandibular Distraction versus Tracheostomy in Neonates with Pierre Robin Sequence Christopher M. Runyan, MD, PhD 1 * , Armando Uribe-Rivera 1 * , Audrey Karlea 2 , Jareen Meinzen-Derr, PhD 3 , Dawn Rothchild 1 , Howard Saal, MD 4 , Robert J. Hopkin, MD 4§ , and Christopher B. Gordon, MD 1§

Surgery Foundation 2014 Reprints and permission:

sagepub.com/journalsPermissions.nav DOI: 10.1177/0194599814542759 http://otojournal.org

Received January 29, 2014; revised May 13, 2014; accepted June 18, 2014.

Sponsorships or competing interests that may be relevant to content are dis- closed at the end of this article.

Abstract Objective . To evaluate costs associated with surgical treat- ment for neonates with Pierre Robin sequence (PRS). Study Design . Retrospective cohort study. Setting . Cincinnati Children’s Hospital Medical Center. Subjects and Methods . With Institutional Review Board approval, we retrospectively studied neonates with PRS treated from 2001 to 2009 with either tracheostomy (Trach), mandibular distraction (MD), or Trach with subsequent MD (Trach 1 MD). Actual charges over a 3-year period associated with operative costs, hospital stay, imaging and sleep studies, clinic visits, and related emergency room visits were col- lected. Home tracheostomy care charges were estimated individually for each patient. Charges were compared using regression and appropriate statistical analyses. Results . Forty-seven neonates were included in the study (MD, n = 26; Trach, n = 12; Trach 1 MD, n = 9). Trach group patients had 2.6-fold higher charges than the MD group despite no difference in length of hospital stay. This difference increased to 7.3-fold when including home trach care-related costs. Trach 1 MD group patients had longer hospital lengths of stay and higher operation room (OR) fees, but no increased total charges compared with the Trach only group. Conclusions . For patients with severe PRS, mandibular distrac- tion provides significant cost savings over tracheostomy ($300,000 per patient over 3 years). Increased costs with tra- cheostomy come from greater hospital-related charges, more frequent airway procedures, a higher incidence of gastrostomy tube feeds, and home trach care costs. A careful examination of long-term outcomes will be critical as mandibular distraction continues to gain acceptance for treatment of PRS. Keywords Pierre Robin sequence, tracheostomy, mandibular distrac- tion osteogenesis

Introduction Defined by the triad of micrognathia, glossoptosis, and airway obstruction, 1 Pierre Robin sequence (PRS) is a morbid and potentially lethal condition among neonates. In PRS, mandibular hypoplasia leads to abnormal cephalad and posterior positioning of the tongue, which frequently results in failure of fusion of the secondary palate, and a U-shaped cleft palate. 2 PRS occurs in 1:8500 live births and may occur in isolate or with a genetic syndrome, most commonly Stickler and Velocardiofacial syndromes, and hemifacial microsomia. 3,4 Clinically, PRS is marked by oxygen desa- turations, apnea, gastroesophageal reflux, feeding difficul- ties, and failure to thrive. Mortality rates associated with PRS range from 0% to 21% (median 4.5%). 5 Mild cases may be managed conservatively, using prone positioning, nasopharyngeal or laryngeal mask airways, or palatal obturators. Moderate to severe cases require surgical inter- vention to relieve or bypass the airway obstruction. Surgery for PRS consists of tongue-lip adhesion (TLA), tracheostomy, or mandibular distraction. Given the 1 Division of Plastic Surgery, Cincinnati Children’s Hospital Medical Center, Cincinnati, Ohio, USA 2 Kaiser Permanente Clinical Genetics Department, Oakland, California, USA 3 Division of Biostatistics and Epidemiology, Cincinnati Children’s Hospital Medical Center, Cincinnati, Ohio, USA 4 Division of Human Genetics, Cincinnati Children’s Hospital Medical Center, Cincinnati, Ohio, USA Presented at the 12 th International Congress on Cleft Lip/Palate and Related Craniofacial Anomalies; May 5-10, 2013; Orlando, Florida. Corresponding Author: Christopher M. Runyan, MD, PhD, Division of Plastic Surgery, Cincinnati Children’s Hospital Medical Center, 200 Albert Sabin Way, Mail Location * These authors contributed equally to this article. § These authors contributed equally to this project.

0461, Cincinnati, OH 45219, USA. Email: chris.runyan@cchmc.org

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