September 2019 HSC Section 1 Congenital and Pediatric Problems

International Journal of Pediatric Otorhinolaryngology 115 (2018) 10–18

E. Cole et al.

symptoms and diagnosis of laryngeal cleft and between presenting symptoms and injection compared with the older groups. It is possible that the greater severity of symptoms in the younger group accelerated the work-up and intervention. Alternately, greater severity of symptoms may have been the reason for earlier presentation. Despite di ff erences in the timing of presentation and intervention, the resolution of symptoms was similar between age groups during the median follow-up of 11 months for all age groups. There were no sig- ni fi cant di ff erences in rates of second injection or need for formal surgical repair. Among those who did require a second injection, there was no signi fi cant di ff erence between age groups in time from fi rst to second injection. Thus, early intervention with injection laryngoplasty is bene fi cial even in the 0 – 3-month age group. It does not increase the need for or delay timing of additional procedures. Presenting symptoms indicative of laryngeal cleft include stridor, choking with feeds, shortness of breath, cough, recurrent respiratory infections, or failure to thrive. Because many children who have lar- yngeal clefts also have other congenital anomalies, it may be di ffi cult to determine to what degree the cleft contributed to the presenting symptoms, especially with less severe type I and II laryngeal clefts [ 9 , 10 ]. Many children with laryngeal clefts have other aerodigestive anomalies and/or neurologic, respiratory or medical comorbidities that can result in similar presenting sign and symptoms, speci fi cally stridor, dysphagia and aspiration. Laryngomalacia was a common comorbidity in our study. Laryngomalacia is associated with stridor, dysphagia and aspiration [ 16 ]. Surgical correction of the anatomic anomalies asso- ciated with laryngomalacia can a ff ect breathing and swallowing [ 16 ]. In this study, a small percentage of children did undergo surgical in- tervention for laryngomalacia and therefore the congruent e ff ect these surgeries cannot be separated from the e ff ects of the injection lar- yngoplasty. In addition to examining outcomes by age, we observed signi fi cant variation in the methods used to evaluate symptoms. While swallow studies, including MBS and FEES, were used as objective measures of injection laryngoplasty outcomes, it is not clear if improvement in swallow study results correlates with resolution of other symptoms. In our study, there was poor correlation between MBS, FEES, and symp- toms during CSE, both pre- and post-injection. Most studies used to validate CSE, MBS and FEES have been in adults whereas agreement between CSE, FEES and MBS in children has not been demonstrated previously. While all studies may have clinical value, the results of these tests cannot be compared directly or used interchangeably. While MBS is considered the gold standard, FEES allows more versatility in feeding positions and modalities and does not subject the patient to potentially harmful ionizing radiation. There are a number of studies showing interrater reliability of FEES in children [ 17 , 18 ], but the few studies that compare FEES directly with MBS are small, with 7 – 30 patients included, and show poor correlation [ 19 , 20 ]. Additionally, prospective studies are needed to better evaluate use of MBS and FEES for assessment of injection laryngoplasty outcomes. Limitations of our study include a single-center population, retro- spective analysis, and variation among providers evaluating presenting and follow-up symptoms. The patient population, while large compared with prior studies on laryngeal cleft injection, was limited to patients from one pediatric hospital, so results may not be directly applicable to other centers. Since the study was retrospective, the clinical decisions to perform injection laryngoplasty were based on clinicians ’ judgment rather than a standardized set of symptoms. Evaluation of symptoms on follow-up visits as well as interpretation of FEES and MBS were also done by health care providers without a standardized form and ab- stracted retrospectively. In particular, the reduction of aspiration with thins found by FEES post-injection, which was not corroborated by MBS or CSE, may be biased as the providers performing FEES are also the

ones who did injection laryngoplasties. Other limitations include the variability in patients who received MBS and/or FEES. The decision to obtain a speci fi c diagnostic modality was clinician dependent rather than standardized.

5. Conclusion

In conclusion, we found that injection laryngoplasty is similarly e ff ective in reducing symptoms for children of di ff erent age groups, including the 0 – 3-month range. Our data, however, call into question methods of evaluation that are used to determine the need for and ef- fi cacy of these injections. A formal prospective study is needed to evaluate the validity of MBS and FEES in patients with laryngeal clefts.

Con fl icts of interest

None.

References

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