Outcomes of Mandibular Distraction Osteogenesis in ...

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Feb 27, 2014 - 11 (8.9). 7 (11.3). 4 (6.6). Stickler syndrome. 4 (3.3). 3 (4.8). 1 (1.6). Othera. 42 (3.1). 23 (3.7) ... orofaciodigital, Cornelia de Lange,. Loeys-Dietz ...
Research

Original Investigation

Outcomes of Mandibular Distraction Osteogenesis in the Treatment of Severe Micrognathia Derek J. Lam, MD, MPH; Meredith E. Tabangin, MPH; Tasneem A. Shikary, MD; Armando Uribe-Rivera, DDS; Jareen K. Meinzen-Derr, PhD; Alessandro de Alarcon, MD, MPH; David A. Billmire, MD; Christopher B. Gordon, MD

IMPORTANCE Patients with severe micrognathia are predisposed to airway obstruction. Mandibular distraction osteogenesis (MDO) is an alternative to tracheotomy that lengthens the mandible in order to improve the retrolingual airway. This study presents outcomes from one of the largest cohorts reported. OBJECTIVE To assess the rate and predictors of surgical success and complications among (1) patients who underwent MDO prior to other airway procedures (MDO first), and (2) patients who required an initial tracheotomy and were subsequently treated with MDO (tracheotomy first). DESIGN, SETTING, AND PARTICIPANTS Retrospective cohort study at a tertiary care pediatric medical center of patients diagnosed as having micrognathia resulting in symptomatic airway obstruction (Pierre Robin sequence) and who underwent MDO from September 1995 to December 2009. INTERVENTIONS Electronic medical records were reviewed. Multivariable regression analysis

was used to assess for predictors of outcome. MAIN OUTCOMES AND MEASURES Rates of surgical success (defined as either tracheotomy avoidance or decannulation) and complications. Potential predictors included demographics, syndrome presence, follow-up time, and surgical history. RESULTS A total of 123 patients (61 in MDO-first subgroup, 62 in tracheotomy-first subgroup) underwent MDO during the study period. Median age at time of distraction was 21 months (range, 7 days–24 years). Surgical success and complication rates were 83.6% and 14.8% in the MDO-first subgroup and 67.7% and 38.7% in the tracheotomy-first subgroup. Tracheotomy-first patients were more likely to have a syndromic diagnosis (66.0% vs 43.0%; P = .009) and were older at the time of MDO (median age, 30 months vs 5.1 months; P < .001). Poorer odds of success were associated with the need for 2 or more other airway procedures (odds ratio [OR], 0.14 [95% CI, 0.02-0.82]) in the MDO-first subgroup and craniofacial microsomia or Goldenhar syndrome (OR, 0.07 [95% CI, 0.009-0.52]) in the tracheotomy-first subgroup. CONCLUSIONS AND RELEVANCE Mandibular distraction osteogenesis has a high rate of success in avoiding tracheotomy. Patients who required a tracheotomy before MDO had a lower success rate in achieving decannulation and a higher rate of complications. However, these patients also had a higher rate of syndromic diagnoses and associated comorbidities. Patients with Goldenhar syndrome have a decreased likelihood of surgical success.

JAMA Otolaryngol Head Neck Surg. 2014;140(4):338-345. doi:10.1001/jamaoto.2014.16 Published online February 27, 2014. 338

Author Affiliations: Department of Otolaryngology–Head and Neck Surgery, Doernbecher Children’s Hospital, Oregon Health and Science University, Portland (Lam); Division of Otolaryngology–Head and Neck Surgery, Cincinnati Children’s Hospital Medical Center, Cincinnati, Ohio (Lam, Shikary, de Alarcon); Division of Biostatistics and Epidemiology, Cincinnati Children’s Hospital Medical Center, Cincinnati, Ohio (Tabangin, Meinzen-Derr); Division of Plastic Surgery, Cincinnati Children’s Hospital Medical Center, Cincinnati, Ohio (Uribe-Rivera, Billmire, Gordon). Corresponding Author: Derek J. Lam, MD, MPH, Department of Otolaryngology–Head and Neck Surgery, Oregon Health and Science University, 3181 SW Sam Jackson Park Rd, PV-01, Portland, OR 97239-3098 ([email protected]). jamaotolaryngology.com

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Mandibular Distraction Osteogenesis

Original Investigation Research

P

ierre Robin sequence is characterized by the triad of micrognathia, glossoptosis, and resultant airway obstruction owing to constriction of the retrolingual space.1 Though not classically described as part of its definition, cleft palate is a commonly associated finding that occurs in up to 90% of children with Pierre Robin sequence. Symptoms of airway obstruction may range from snoring and stertor while asleep to frank obstruction and retractions when awake. Gasping or aspiration while feeding is frequently encountered, and this in combination with chronic airway obstruction can cause failure to thrive. Long-term sequelae of severe airway obstruction can lead to cor pulmonale and cardiorespiratory arrest. Interventions aimed at relieving such symptoms vary depending on the severity of the symptoms. Conservative measures for less severe symptoms include prone positioning and use of a nasopharyngeal airway.2 In the event that such conservative treatments are unsuccessful or for more severe symptoms, options for surgical intervention include tongue-lip adhesion, tracheotomy, and mandibular distraction osteogenesis (MDO). Tongue-lip adhesion has been shown to significantly improve obstructive sleep apnea due to micrognathia3 but has generally been less effective than MDO at normalizing obstructive symptoms, particularly in severely symptomatic patients. In addition, tongue-lip adhesion can lead to dysphagia and feeding difficulties.4 Tracheotomy offers a definitive treatment for upper airway obstruction but has associated risks of accidental decannulation or mucous plugging. There is also potential long-term morbidity related to peristomal scarring and tracheal erosion in addition to the need for long-term maintenance and home care.5,6 In recent years, there have been an increasing number of reports on the results of MDO as an alternative to tracheotomy.7-15 The goals of this study were to assess the surgical success and complication rates of MDO for treatment of severe micrognathia and to identify potential predictors of surgical success and complications. Surgical success was defined as either (1) avoidance of tracheotomy or (2) decannulation among those patients treated initially with a tracheotomy.

Methods Participants This was a retrospective cohort study of all patients who underwent MDO from September 1, 1995, to December 31, 2009, at Cincinnati Children’s Hospital Medical Center. Inclusion criteria included any patients who underwent initial MDO during the study period. All patients were seen through a multidisciplinary craniofacial clinic, and those with a concern for syndromic Pierre Robin sequence were routinely evaluated by clinicians from both the genetics and ophthalmology departments in addition to the craniofacial surgery and otolaryngology departments to ensure proper diagnosis and management. Electronic and paper chart medical records were reviewed for relevant data. Patients who were lost to follow-up after MDO or had incomplete medical records were excluded. Mandibular distraction osteogenesis was performed with short sagittal split osteotomies using primarily external jamaotolaryngology.com

distraction devices, although internal distraction devices were used in a minority of patients based on surgeon preference. This study was approved by the institutional review board of Cincinnati Children’s Hospital.

Potential Predictors Variables included as potential predictors of outcome included demographics (sex, age at time of distraction), follow-up time, syndrome presence (categorized as isolated Pierre Robin sequence, craniofacial microsomia [CFM] or Goldenhar syndrome, Treacher-Collins syndrome, and other syndromes), type of initial surgical intervention (tracheotomy vs MDO), length of mandible distracted, number of distractions, and number of subsequent airway procedures (eg, laryngotracheoplasty, endoscopic airway procedures, base of tongue procedures, choanal atresia repair).

Outcomes While the primary goal of MDO was to improve the retrolingual airway and relieve airway obstruction, the definition of surgical success necessarily differed for patients who were initially treated with tracheotomy prior to MDO compared with those who underwent MDO as an initial procedure. Thus, surgical success was defined as (1) avoidance of tracheotomy among patients who were treated first with MDO and (2) successful decannulation among patients who initially underwent tracheotomy prior to MDO. Complications assessed included open bite deformity, premature bone consolidation, temporomandibular joint (TMJ) ankylosis, facial nerve injury, emergent reintubation, and prolonged intubation. For the purposes of this analysis, the need for repeated distraction more than 30 days after the initial distraction was not considered a complication because it was felt that this need reflected a lack of innate growth of the distracted mandible over time rather than a failure of the initial distraction.

Statistical Analysis Descriptive statistics are reported as means (SDs) and medians with interquartile ranges (IQRs) or frequencies with percentages. The characteristics of the 2 subgroups defined by initial surgical treatment were compared using the Wilcoxon rank sum test for continuous variables and the χ2 or Fisher exact test for categorical variables. Logistic regression analysis was used to assess the relationship between the potential predictors and each dichotomous outcome of interest (surgical success and occurrence of complications). The analysis of surgical success was stratified by initial surgical intervention (tracheotomy vs MDO), since the definition of outcome differed between these subgroups. For potential predictors of a complication, all complications were grouped together as a dichotomous outcome (any complication or no complication). The entire cohort was included in this regression analysis since the definition of a complication was the same regardless of the initial intervention. For each outcome (surgical success and occurrence of a complication), logistic regression models were constructed to identify potentially important associations between the predictor variables and each outcome of interest. A conservative criterion P = .20 was used as a cutoff for inclu-

JAMA Otolaryngology–Head & Neck Surgery April 2014 Volume 140, Number 4

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339

Research Original Investigation

Mandibular Distraction Osteogenesis

Table 1. Characteristics and Outcomes of 123 Patients in Cohort Characteristic Male

No. (%)

Tracheotomy First (n = 62 [50.4%])

MDO First (n = 61 [49.6%])

Isolated Pierre Robin sequence

54 (43.9)

21 (33.9)

35 (57.4)

Syndromic

69 (56.1)

41 (66.3)

26 (42.6)

Treacher-Collins syndrome

12 (9.8)

8 (12.9)

4 (6.6)

CFM–Goldenhar syndrome

11 (8.9)

7 (11.3)

4 (6.6)

4 (3.3)

3 (4.8)

1 (1.6)

42 (3.1)

23 (3.7)

17 (2.8)

Mean (SD)

40 (52)

46 (47)

34 (57)

Median (IQR)

21 (2.2-48.2)

30 (15.2-52.8)

5.1 (0.6-42.1)

Stickler syndrome Othera

P Value

69 (56.1)

.009

Age at distraction, mo

Range Follow-up time, mean (SD), y Amount distracted, mean (SD), mm

6 d–20 y

25 d–20 y

.20