Research
Original Investigation
Pediatric Vocal Fold Immobility Natural History and the Need for Long-term Follow-up Jad Jabbour, MD, MPH; Timothy Martin, MD; David Beste, MD; Thomas Robey, MD
IMPORTANCE The clinical course and outcomes of pediatric vocal fold immobility (VFI) vary widely in the literature, and follow-up in these patients varies accordingly. A better understanding of the natural history of pediatric VFI is crucial to improved management. OBJECTIVE To characterize the natural history of pediatric VFI, including symptoms and rates of resolution and surgical intervention. DESIGN, SETTING, AND PARTICIPANTS Retrospective review at an academically affiliated private pediatric otolaryngology practice in a metropolitan area of all patients seen between July 15, 2001, and September 1, 2012, with a diagnosis of complete or partial VFI. After elimination of 92 incomplete or duplicate files, 404 patient records were reviewed for demographic characteristics, etiologies, symptoms, follow-up, resolution, and interventions. Follow-up records were available for 362 patients (89.6%). MAIN OUTCOMES AND MEASURES Resolution of VFI confirmed by repeated laryngoscopy, length of follow-up, and surgical intervention rates. RESULTS Among the 404 patients, left VFI was present in 66.8%, right VFI in 7.9%, and bilateral VFI in 25.3%. Median (range) age at presentation was 2.9 (0-528.1) months. Major etiological categories included cardiac surgery in 68.8%, idiopathic immobility in 21.0%, and neurologic disease in 7.4%. At presentation, 61.4% experienced dysphonia, 54.0% respiratory symptoms, and 49.5% dysphagia. Tracheotomy was performed in 25.7% and gastrostomy in 40.8%. Median (range) duration of follow-up among the 89.6% of patients with follow-up was 17.2 (0.2-173.5) months. Resolution evidenced by laryngoscopy was found in 28.0%, with a median (range) time to resolution of 4.3 (0.4-38.7) months. In patients without laryngoscopic resolution, median follow-up was 26.0 months, and 28.9% reported symptomatic resolution. CONCLUSIONS AND RELEVANCE The natural history of pediatric VFI involves substantial morbidity, with lasting symptoms and considerable rates of surgical intervention. In this large database, the majority of patients did not experience resolution. This suggests a need for more regimented follow-up in these patients, a recommendation for which is proposed here.
Author Affiliations: Department of Otolaryngology and Communication Sciences, Medical College of Wisconsin, Milwaukee (Jabbour, Martin, Beste, Robey); Division of Pediatric Otolaryngology, Children’s Hospital of Wisconsin, Milwaukee (Martin, Beste, Robey).
JAMA Otolaryngol Head Neck Surg. 2014;140(5):428-433. doi:10.1001/jamaoto.2014.81 Published online March 13, 2014. 428
Corresponding Author: Thomas Robey, MD, Department of Otolaryngology and Communication Sciences, Medical College of Wisconsin, 9000 W Wisconsin Ave, Ste 340, Milwaukee, WI 53201 ([email protected]). jamaotolaryngology.com
Copyright 2014 American Medical Association. All rights reserved.
Downloaded From: http://archotol.jamanetwork.com/ by a Georgetown University Medical Center User on 05/25/2015
Pediatric Vocal Fold Immobility
Original Investigation Research
V
ocal fold immobility (VFI) in children is related to a variety of causes, including cardiac surgical procedures, neurologic conditions, and idiopathic injury.1 Complications caused by VFI include respiratory problems, voice alterations, and feeding difficulties, the distribution of which depends on whether a patient has bilateral or unilateral involvement. Despite what is known about pediatric VFI, important questions remain. Reported recovery rates range widely in the literature, from 8% to 82%2-7; accordingly, follow-up in these patients is also quite variable, with no established guidelines or recommendations. As children with VFI live longer as a result of advancements in pediatric cardiac surgery, as well as improved management of certain neurologic conditions, the aforementioned sequelae have the potential to substantially affect a child’s life. The possibility of therapeutic intervention in these children has also expanded, including options for injection laryngoplasty, thyroplasty, and nerve reinnervation.8-11 The pediatric otolaryngologist has become a key caregiver for patients who do not recover their vocal fold function and manages these issues into adulthood. Yet this management—as well as counseling at the time of diagnosis—is often challenging given the questions that remain regarding the natural history of pediatric VFI and the appropriate follow-up in these patients. This article presents, to our knowledge, the largest series of pediatric patients with VFI reported in the literature. The purpose of our retrospective review was to better characterize the natural history of pediatric VFI. We highlight the common etiologies and manifestations of VFI, the presenting symptoms and the comorbidities often associated with VFI, and the resolution rate and surgical intervention rate for these children. For those patients who do not recover, the issues of ongoing symptoms and the challenges of consistent follow-up are also discussed. A recommended follow-up schedule is proposed.
Methods This study was a retrospective review of medical records of patients at a private pediatric otolaryngology practice in Milwaukee, Wisconsin. The practice was affiliated with the Children’s Hospital of Wisconsin, and the study was approved by that hospital’s institutional review board. Consent was waived due to the nature of the study. All patients seen between July 15, 2001, and September 1, 2012, with a diagnosis of unilateral or bilateral, partial or complete “Paralysis of Vocal Cords or Larynx” (International Classification of Diseases, Ninth Revision, Clinical Modification diagnosis codes 478.31-478.35) were included. For the purposes of this study, we combined all of these patients under the heading of VFI. All diagnoses were made by means of flexible laryngoscopy by 1 of 3 attending pediatric otolaryngologists (T.M., D.B., T.R.) in the clinic or in consultation at 1 of 3 area hospitals. A total of 496 patient files were identified. Patient records were eliminated from the database for the following reasons: insufficient information regarding diagnosis, duplicate files, or failure to confirm VFI when laryngoscopy was repeated within 1 week of diagnosis. A total of 404 records rejamaotolaryngology.com
Table 1. General Characteristics of Total Study Population, Including Laterality and Etiology of VFI Characteristic
No. (%) (N = 404)
Immobility Left Right Bilateral
270 (66.8) 32 (7.9) 102 (25.2)
Etiology Cardiac surgery
278 (68.8)
Idiopathic
85 (21.0)
Neurologic
30 (7.4)
Mixeda
5 (1.2)
Miscellaneousb
6 (1.5)
Sex Male
211 (52.2)
Female
193 (47.8)
Age at diagnosis Mean, mo Median (range), mo
21.6 2.9 (0-528.1)
Original Investigation
Pediatric Vocal Fold Immobility Natural History and the Need for Long-term Follow-up Jad Jabbour, MD, MPH; Timothy Martin, MD; David Beste, MD; Thomas Robey, MD
IMPORTANCE The clinical course and outcomes of pediatric vocal fold immobility (VFI) vary widely in the literature, and follow-up in these patients varies accordingly. A better understanding of the natural history of pediatric VFI is crucial to improved management. OBJECTIVE To characterize the natural history of pediatric VFI, including symptoms and rates of resolution and surgical intervention. DESIGN, SETTING, AND PARTICIPANTS Retrospective review at an academically affiliated private pediatric otolaryngology practice in a metropolitan area of all patients seen between July 15, 2001, and September 1, 2012, with a diagnosis of complete or partial VFI. After elimination of 92 incomplete or duplicate files, 404 patient records were reviewed for demographic characteristics, etiologies, symptoms, follow-up, resolution, and interventions. Follow-up records were available for 362 patients (89.6%). MAIN OUTCOMES AND MEASURES Resolution of VFI confirmed by repeated laryngoscopy, length of follow-up, and surgical intervention rates. RESULTS Among the 404 patients, left VFI was present in 66.8%, right VFI in 7.9%, and bilateral VFI in 25.3%. Median (range) age at presentation was 2.9 (0-528.1) months. Major etiological categories included cardiac surgery in 68.8%, idiopathic immobility in 21.0%, and neurologic disease in 7.4%. At presentation, 61.4% experienced dysphonia, 54.0% respiratory symptoms, and 49.5% dysphagia. Tracheotomy was performed in 25.7% and gastrostomy in 40.8%. Median (range) duration of follow-up among the 89.6% of patients with follow-up was 17.2 (0.2-173.5) months. Resolution evidenced by laryngoscopy was found in 28.0%, with a median (range) time to resolution of 4.3 (0.4-38.7) months. In patients without laryngoscopic resolution, median follow-up was 26.0 months, and 28.9% reported symptomatic resolution. CONCLUSIONS AND RELEVANCE The natural history of pediatric VFI involves substantial morbidity, with lasting symptoms and considerable rates of surgical intervention. In this large database, the majority of patients did not experience resolution. This suggests a need for more regimented follow-up in these patients, a recommendation for which is proposed here.
Author Affiliations: Department of Otolaryngology and Communication Sciences, Medical College of Wisconsin, Milwaukee (Jabbour, Martin, Beste, Robey); Division of Pediatric Otolaryngology, Children’s Hospital of Wisconsin, Milwaukee (Martin, Beste, Robey).
JAMA Otolaryngol Head Neck Surg. 2014;140(5):428-433. doi:10.1001/jamaoto.2014.81 Published online March 13, 2014. 428
Corresponding Author: Thomas Robey, MD, Department of Otolaryngology and Communication Sciences, Medical College of Wisconsin, 9000 W Wisconsin Ave, Ste 340, Milwaukee, WI 53201 ([email protected]). jamaotolaryngology.com
Copyright 2014 American Medical Association. All rights reserved.
Downloaded From: http://archotol.jamanetwork.com/ by a Georgetown University Medical Center User on 05/25/2015
Pediatric Vocal Fold Immobility
Original Investigation Research
V
ocal fold immobility (VFI) in children is related to a variety of causes, including cardiac surgical procedures, neurologic conditions, and idiopathic injury.1 Complications caused by VFI include respiratory problems, voice alterations, and feeding difficulties, the distribution of which depends on whether a patient has bilateral or unilateral involvement. Despite what is known about pediatric VFI, important questions remain. Reported recovery rates range widely in the literature, from 8% to 82%2-7; accordingly, follow-up in these patients is also quite variable, with no established guidelines or recommendations. As children with VFI live longer as a result of advancements in pediatric cardiac surgery, as well as improved management of certain neurologic conditions, the aforementioned sequelae have the potential to substantially affect a child’s life. The possibility of therapeutic intervention in these children has also expanded, including options for injection laryngoplasty, thyroplasty, and nerve reinnervation.8-11 The pediatric otolaryngologist has become a key caregiver for patients who do not recover their vocal fold function and manages these issues into adulthood. Yet this management—as well as counseling at the time of diagnosis—is often challenging given the questions that remain regarding the natural history of pediatric VFI and the appropriate follow-up in these patients. This article presents, to our knowledge, the largest series of pediatric patients with VFI reported in the literature. The purpose of our retrospective review was to better characterize the natural history of pediatric VFI. We highlight the common etiologies and manifestations of VFI, the presenting symptoms and the comorbidities often associated with VFI, and the resolution rate and surgical intervention rate for these children. For those patients who do not recover, the issues of ongoing symptoms and the challenges of consistent follow-up are also discussed. A recommended follow-up schedule is proposed.
Methods This study was a retrospective review of medical records of patients at a private pediatric otolaryngology practice in Milwaukee, Wisconsin. The practice was affiliated with the Children’s Hospital of Wisconsin, and the study was approved by that hospital’s institutional review board. Consent was waived due to the nature of the study. All patients seen between July 15, 2001, and September 1, 2012, with a diagnosis of unilateral or bilateral, partial or complete “Paralysis of Vocal Cords or Larynx” (International Classification of Diseases, Ninth Revision, Clinical Modification diagnosis codes 478.31-478.35) were included. For the purposes of this study, we combined all of these patients under the heading of VFI. All diagnoses were made by means of flexible laryngoscopy by 1 of 3 attending pediatric otolaryngologists (T.M., D.B., T.R.) in the clinic or in consultation at 1 of 3 area hospitals. A total of 496 patient files were identified. Patient records were eliminated from the database for the following reasons: insufficient information regarding diagnosis, duplicate files, or failure to confirm VFI when laryngoscopy was repeated within 1 week of diagnosis. A total of 404 records rejamaotolaryngology.com
Table 1. General Characteristics of Total Study Population, Including Laterality and Etiology of VFI Characteristic
No. (%) (N = 404)
Immobility Left Right Bilateral
270 (66.8) 32 (7.9) 102 (25.2)
Etiology Cardiac surgery
278 (68.8)
Idiopathic
85 (21.0)
Neurologic
30 (7.4)
Mixeda
5 (1.2)
Miscellaneousb
6 (1.5)
Sex Male
211 (52.2)
Female
193 (47.8)
Age at diagnosis Mean, mo Median (range), mo
21.6 2.9 (0-528.1)
