2015 HSC Section 1 Book of Articles

Runyan et al

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MD Trach Trach+MD

Non-syndromic Syndromic

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buildup of airway granulation tissue, and occasional need for complicated revision surgery, including laryngotracheoplasty and cricotracheal resection. 11-14 Further, patients with a tra- cheostomy typically require multiple ML&B procedures to investigate these morbidities and to prepare for decannula- tion. Of greatest significance, tracheostomy is associated with a small but real chance of mortality (1%-5%). 15 Mandibular distraction differs in that it directly addresses the primary problem, micrognathia. Using MD to lengthen the mandible provides greater room for the tongue and oral soft tissues and indirectly pulls them forward by their attach- ments to the mandible, correcting glossoptosis and improving airway obstruction. A growing body of studies indicate MD helps PRS patients treated with tracheostomy achieve decan- nulation sooner or avoid tracheostomy altogether. 9,11,14,16-24 Complications associated with MD include hardware mal- function, infection, damage to tooth buds, and nerve injury and pain, although the actual incidence varies depending on surgeon experience and technique. 25,26 Two groups have performed cost analyses to compare tracheostomy to MD for PRS. Kohan et al 27 examined 149 neonates with PRS treated with either internal MD (n = 43) or tracheostomy (n = 73). They reported a 2-fold higher cost for the Trach group ($382,246) compared to MD group ($193,128) over a 4-year follow-up period. The cost differ- ence was due to an increased length of ICU stay in patients receiving tracheostomy. Hong et al 28 examined 52 patients with PRS: 21 received MD, and 31 had a tracheostomy. With 1 year of follow-up data, the Trach group had a 1.6- fold increase in cost compared to the MD group ($92,164 vs $57,649, Canadian dollars). This cost difference was attrib- uted to increased hospital stay for tracheostomy patients, as their health system mandates 90 days in house for home tra- cheostomy care arrangement. Both studies used averaged operative and ICU per diem fees rather than individual patients’ billed charges, as done in our study. Also, neither Year 1 Year 2 Year 3 Figure 4. Annual charges ($USD) of syndromic versus non-syn- dromic patients. Cost comparison (not including home care charges) of patients with non-syndromic versus syndromic Pierre Robin sequence over 3-year period. Median values (horizontal line) presented with twenty-fifth through seventy-fifth percentile ranges. Wilcoxon rank sum tests used to compare the 2 groups at each time point: Year 1: P = .26; Year 2: P = .03; Year 3: P = .13.

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hospital charges to identify total charges for the groups ( Figure 3 ). As expected, the addition of home health care resulted in a greater disparity between patients receiving a tracheostomy versus those receiving MD only. We observed that the Trach 1 MD group incurred fewer charges than the Trach group in all 3 years, although this difference was not statistically significant (Year 1 P = .27, Year 2 P = .30, Year 3 P = .29). This trend may be attributed to a higher rate of decannulation among Trach 1 MD (5 of 9, 56%) versus Trach (2 of 12, 17%) over the 3-year period. To examine the contribution of syndromic status, all patients were grouped based on diagnosis of isolated PRS (34 patients, 72%) or syndromic (including unknown syn- dromes) PRS (13 patients, 28%). As shown in Table 1 , although the MD group had a higher percentage of nonsyn- dromic patients, this difference was not significant ( P = .10). Figure 4 compares the charges between these 2 groups. Syndromic patients had higher associated charged for all 3 years; however, this was only significant during year 2 ( P = .03). Discussion Tracheostomy effectively bypasses tongue-based obstruction and remains the gold standard for severe obstruction that may occur with PRS. However, tracheostomy has greater associated morbidity including negative long-term speech effects, difficulties with feeding, psychosocial delays, fre- quent hospital admissions for tracheitis and pneumonia, Year 3 Figure 3. Estimated total charges ($USD) of groups inclusive of home health care costs. Total charges over 3 years following initial surgical intervention for patients with Pierre Robin sequence receiving mandibular distraction (MD), tracheostomy (Trach), or tracheostomy with subsequent MD (Trach 1 MD), including home tracheostomy-care costs (eg, supplies, equipment rental, and home nursing fees). Median values (horizontal line) are presented with twenty-fifth through seventy-fifth percentile ranges. Statistical analy- ses by year: years 1-3: P \ .01 (Kruskal-Wallis comparison for non- parametric data). A Wilcoxon rank sum test was used to compare Trach vs Trach 1 MD groups at each time point: year 1: P = .27; year 2: P = .30; year 3: P = .29. Year 1 Year 2

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