The Laryngoscope C 2014 The American Laryngological, V
Rhinological and Otological Society, Inc.
Idiopathic Bilateral Vocal Cord Paralysis in Infants: Case Series and Literature Review Maria Lesnik, MD; Briac Thierry, MD; Marion Blanchard, MD; Fergal Glynn, MD, FRCS; Franc¸oise Denoyelle, MD, PhD; Vincent Couloigner, MD, PhD; No€el Garabedian, MD; Nicolas Leboulanger, MD, PhD Objectives/Hypothesis: Vocal fold paralysis is the second most common congenital laryngeal anomaly in newborns. Bilateral paralysis is a severe condition and often remains of unknown etiology. We report our experience of congenital idiopathic bilateral vocal fold paralysis in newborns and infants, and discuss the therapeutic options. Study Design: Retrospective review. Methods: A retrospective review was carried out at a single tertiary referral center over a 15-year period of children presenting with congenital idiopathic bilateral laryngeal paralysis (CIBP). Results: Twenty-six patients were identified and managed over period of the study (mean follow-up, 6.7 years). A tracheostomy was eventually performed in 14 patients, 12 during the neonatal period. Endoscopic treatment was performed in 16 patients and an external approach in five patients after endoscopic failure. Three patients were managed by watchful waiting. Spontaneous recovery occurred in 16 patients (median age, 14.5 months), including nine with a tracheostomy. Decannulation was achieved in 12 patients (median age, 42 months), and noninvasive positive pressure ventilation (NPPV) was applied in six patients. Conclusions: Only inhomogeneous series of pediatric bilateral laryngeal palsies have been published so far, making it difficult to identify prognostic and predictive markers, as well as therapeutic guidelines. Focusing on CIBP, our data suggest that the management strategy must take into account the possibility of a delayed spontaneous recovery. NPPV is an interesting therapeutic tool in this situation. A systematic somatic assessment is mandatory to look for associated conditions. Key Words: Bilateral vocal fold paralysis, congenital, idiopathic, tracheotomy, noninvasive positive pressure ventilation. Level of Evidence: 4 Laryngoscope, 00:000–000, 2014
INTRODUCTION Bilateral vocal fold paralysis is a cause of stridor and respiratory distress in neonates and infants. Early diagnosis can be made by fiberoptic examination of the larynx, but a significant proportion of the children will need immediate intubation after birth because of intense respiratory distress. Bilateral paralysis represents from 30% to 60% of all laryngeal anomalies in children.1–5 Its etiologies may be traumatic (delivery, surgery), neurological, and idiopathic, with an estimated incidence of 40%, 40%, and 30%, respectively.6 Traumatic etiology of a bilateral laryngeal palsy is commonly an easy diagnosis, and a neurologically associated condition can usually be
From the Department of Pediatric Otolaryngology–Head and Neck Surgery, Necker Enfants-Malades Hospital, Paris, France Editor’s Note: This Manuscript was accepted for publication November 10, 2014. Presented at the 2013 meeting of the French Otolaryngology Society (SFORL), Paris, France, October 2013, and the SIFORL Meeting, Montreal, Canada, June 2014. The authors have no funding, financial relationships, or conflicts of interest to disclose. Send correspondence to Nicolas Leboulanger, MD, Department of Otolaryngology–Head and Neck Surgery, Necker Enfants Malades Hospital, 149 rue de Se`vres, 75015 Paris, France. E-mail: nicolas.leboulanger@ nck.aphp.fr DOI: 10.1002/lary.25076
Laryngoscope 00: Month 2014
identified by additional explorations. When no specific cause is found, the condition is considered idiopathic. Management options in congenital bilateral laryngeal paralysis extend from watchful waiting to early tracheostomy. Several therapeutic options are possible including numerous surgical procedures. The possibility of a spontaneous recovery must always be kept in mind. Most of the reports published in the international literature so far address both unilateral and bilateral paralysis and encompass several different etiologies. To our knowledge, no series of patients has specifically focused on the evolution, management, and outcomes of congenital idiopathic bilateral laryngeal paralysis (CIBP). Therefore, the aim of this study was to report a series of pediatric patients presenting only with CIBP to better define the prognosis of this specific condition and analyze its functional outcome with regard to management strategy, decannulation rates for children who required a tracheostomy, restoration of the airway, dysphonia, and swallowing function.
MATERIALS AND METHODS We performed a retrospective review over a 15-year period (1998–2013) of all children treated in our institution for CIBP. In all patients, the diagnosis was eventually made by fiberoptic examination of the larynx. An endoscopic examination
Lesnik et al.: Idiopathic Bilateral Laryngeal Palsy
1
TABLE I. Patients. Patient
Sex
Age at Diagnosis, d
Initial Symptoms
Preterm?
Associated Condition
Intubation at Birth?
Duration, d
1
F
3
2 3
F F
60 120
4
F
7
Respiratory distress
No
Severe GERD
Yes
60
5 6
F F
21 0
Respiratory distress Stridor 1 dysphonia
No No
Severe GERD
Yes No
21
7
M
1,100
Respiratory distress
Yes
HMD
Yes
60
8 9
F M
480 1,825
Respiratory distress Respiratory distress
Yes Yes
BPD, PDA HMD, BPD, autism
Yes Yes
90 120
10
F
3
Respiratory distress
No
Yes
30
11 12
F M
21 2
Respiratory distress Stridor
No No
21
Esophageal atresia
Yes No
13
M
60
Respiratory distress
No
RDS
Yes
60
14 15
M M
15 0
Dyspnea at exertion Stridor
No No
16
F
0
Stridor
Yes
17 18
F F
0 8
Stridor Stridor
Yes Yes
19
F
60
Respiratory distress
No
Yes
60
20 21
M F
7 0
Respiratory distress Respiratory distress
No No
Yes Yes
60 21
22
M
90
Respiratory distress
Yes
Yes
180
23 24
F F
0 45
Respiratory distress Respiratory distress
No No
Yes Yes
180 10
25
M
120
Respiratory distress
Yes
Yes
10
26
M
540
Respiratory distress
Yes
Yes
?
Stridor
No
No
—
Stridor Respiratory distress
No No
No Yes
— 8
No Yes
3
HMD
Yes
90
HMD and BPD
No No
HMD
HMD
BPD 5 bronchopulmonary dysplasia; F 5 female; GERD 5 gastroesophageal reflux disease; HMD 5 hyaline membrane disease; M 5 male; PDA 5 patent ductus arteriosus; RDS 5 respiratory distress syndrome.
of the airway was also performed in all patients looking for associated airway malformations. To confirm the idiopathic type of the paralysis and assess its clinical impact, the following exams were systematically performed in each patient, during the first months of life for newborns and neonates, and after the diagnosis was made for the few late-diagnosed patients: neuropediatric clinical examination, cardiac ultrasonography, cerebral magnetic resonance imaging (MRI) (after 4 months of age), esophageal pH test, chest radiography, and barium swallow. All children presenting with a postoperative or an obstetrical (following dystocia) bilateral vocal fold paralysis were excluded. Our institutional ethics committee approved this study.
RESULTS During the considered period, 154 children were diagnosed with a laryngeal paralysis. Among them, 26 presented with a CIBP and were included in the study, which included 16 females and 10 males.
Patients The diagnosis of bilateral laryngeal palsy was made by fiberoptic examination of the larynx at a median age Laryngoscope 00: Month 2014
2
of 18 days in the intensive care unit or in the operating room for patients intubated at birth. The mean age at diagnosis would not be representative of the series because of some patients (patients 7, 8, 9, 26) who were diagnosed with a considerable delay. Nineteen patients (73%) required tracheal intubation at birth, for a mean duration of 60 days (range, 3– 180 days). Comorbidities were found in 11 patients, but none known to be responsible for a bilateral laryngeal paralysis. Nineteen children presented eventually with failure to thrive and required temporary feeding by nasogastric tube. No patients required a gastrostomy tube. Patients’ data are summarized in Tables I and II.
Initial Management An endoscopic assessment under local followed by general anesthesia was performed in all patients after 2 months (median) of age. Eleven patients displayed a CIBP in full adduction and 15 in subtotal adduction. No patients had a CIBP in abduction. Some patients presented minor anomalies of the airway (narrow cricoid and subglottis, n 5 4; suprastomal granuloma, n 5 7), but not sufficient enough to significantly reduce the airway. Lesnik et al.: Idiopathic Bilateral Laryngeal Palsy
TABLE II. Management. Initial Management
Secondary Management Age at Tracheotomy, d
Extubation failure?
Tracheotomy?
1
No
2 3
No No
4
Yes
Yes
5 6
Yes No
Yes No
7
Yes
No
No
BC, cricoid split
2
8 9
No No
No Yes
2,000
No No
BC, cricoid split BC, arytenoidectomy
2 4
10
Yes
Yes
33
No
BC, arytenoidectomy
4
Yes
11 12
Yes Yes
Yes Yes
21 60
No No
BC, arytenoidectomy UC
3 1
No Yes
13
Yes
Yes
50
14 15
No No
No No
16
Yes
Yes
17 18
No No
No No
19
Yes
Yes
58
No
UC, arytenoidectomy
3
Laryngoplasty
2
No
20 21
Yes Yes
Yes Yes
65 19
No No
BC, arytenoidectomy
5
Laryngoplasty
1
Yes No
22
Yes
Yes
180
No
23 24
Yes No
Yes No
190
No No
UC, arytenoidectomy BC
2 2
Arytenoidopexy
1
No
BC
3 Laryngoplasty
4
Patient
Watchful Waiting?
Endoscopic Procedure
No.
No
No
UC, arytenoidectomy
2
No
No No
No Yes
UC, arytenoidectomy
1
No No
66
No
UC, arytenoidectomy
2
15
No Yes
25
No
No
26
Yes
Yes
100
External Approach
No.
No Yes No No Laryngoplasty Laryngoplasty
1 1
No No
No
BC, arytenoidectomy
7
No
No Yes
BC
2
No No
No
UC, arytenoidectomy
1
No
Yes Yes
630
NPPV
Yes Yes
Yes
No
No No No Yes
BC 5 bilateral cordotomy; NPPV 5 noninvasive positive pressure ventilation; UC 5 unilateral cordotomy.
In all patients, the CIPB was found to be the only cause of upper airway obstruction. All patients eventually underwent the series of examinations described above, confirming the diagnosis of CIBP. All patients were also systematically treated for gastroesophageal reflux by proton pump inhibitors. Among the 19 patients intubated at birth, 14 (74%) could not be extubated and therefore underwent a tracheostomy at a median age of 62 days.
Secondary Management An endoscopic cordotomy was performed in 17 patients: unilateral in seven patients, including three with a tracheostomy; bilateral in 10 patients, including six with a tracheostomy. Mean age at cordotomy was 25 months. Among those 17 patients, 10 (nine with a tracheostomy) secondarily needed an endoscopic arytenoidectomy at a mean age of 33.8 months. None of our patients had a posterior cricoid split with cartilage graft. Fourteen patients underwent at least two endoscopic procedures, and six patients needed a complemenLaryngoscope 00: Month 2014
tary surgery by external approach. Procedures are detailed in Table II. Noninvasive positive pressure ventilation (NPPV) by continuous airway pressure at sleep was initiated in eight patients, including seven with a tracheostomy. In these patients, the NPPV aimed to facilitate the decannulation. No NPPV was initiated in a neonate or for pulmonary reasons.
RESULTS The mean follow-up duration was 6.7 years (range, 2–15 years). Spontaneous recovery occurred in 17 patients (65%), including nine with a tracheostomy, at a mean age of 25 months. Among the 14 patients with a tracheostomy, 12 (85%) were eventually weaned from the cannula, at a mean age of 36 months. Four patients still have NPPV at sleep. Results are summarized in Table III.
DISCUSSION This retrospective study reports on 26 patients presenting with CIBP with regard to the outcomes and Lesnik et al.: Idiopathic Bilateral Laryngeal Palsy
3
TABLE III. Results.
Patient Tracheotomy? NPPV
Weaning From NPPV?
Age at Spontaneous Spontaneous Recovery? Recovery, m Decannulation?
Age at Decannulation, m
Current Clinical Status
Actual Follow-up, y
Stridor at exertion
2.5
1
No
No
Non
2
No
No
Yes
10
Dysphonia
3
3 4
No Yes
No No
Yes Yes
12 12
Intermittent stridor Yes
31
3.25 5
5
Yes
Yes
Yes
15
Yes
30
6 7
No No
No No
Yes Yes
8 48
Excellent
7 10
21
4.5 years old
8
8
No
No
Yes
9 10
Yes Yes
No Yes
No No
11
Yes
No
Yes
12
No
12 13
Yes Yes
Yes No
Yes Yes
20 84
Yes Yes
14
No
No
Yes
15 16
No Yes
No No
Yes No
17
No
Yes
No
NPPV
1
18 19
No Yes
Yes No
Yes No
NPPV
7 4
20
Yes
Yes
21 22
Yes Yes
No Yes
23
Yes
No
No
24 25
No No
No No
Yes No
26
Yes
Yes
No
8 years old
3.6 No Yes
60 11 26
Tracheotomy NPPV
10 5
Cannula capped all day
6.5
Decannulated, NPPC
2.5 8
Stridor at exertion
16
24 No
5 5.5
54
Yes
42
Yes
72
Yes
72
NPPV
8
Yes Yes
3 21
Yes Yes
8 23
Excellent NPPV
1 8
Yes
192
Dysphonia
17
Excellent
2.3 15
NPPV
10
14 Yes
84
NPPV 5 noninvasive positive pressure ventilation.
decannulation rates. The overall results were acceptable for children who needed a tracheostomy, with a decannulation rate of 85%. CIBP is an uncommon condition in pediatric otolaryngology, and its exact incidence is not known but is probably
Rhinological and Otological Society, Inc.
Idiopathic Bilateral Vocal Cord Paralysis in Infants: Case Series and Literature Review Maria Lesnik, MD; Briac Thierry, MD; Marion Blanchard, MD; Fergal Glynn, MD, FRCS; Franc¸oise Denoyelle, MD, PhD; Vincent Couloigner, MD, PhD; No€el Garabedian, MD; Nicolas Leboulanger, MD, PhD Objectives/Hypothesis: Vocal fold paralysis is the second most common congenital laryngeal anomaly in newborns. Bilateral paralysis is a severe condition and often remains of unknown etiology. We report our experience of congenital idiopathic bilateral vocal fold paralysis in newborns and infants, and discuss the therapeutic options. Study Design: Retrospective review. Methods: A retrospective review was carried out at a single tertiary referral center over a 15-year period of children presenting with congenital idiopathic bilateral laryngeal paralysis (CIBP). Results: Twenty-six patients were identified and managed over period of the study (mean follow-up, 6.7 years). A tracheostomy was eventually performed in 14 patients, 12 during the neonatal period. Endoscopic treatment was performed in 16 patients and an external approach in five patients after endoscopic failure. Three patients were managed by watchful waiting. Spontaneous recovery occurred in 16 patients (median age, 14.5 months), including nine with a tracheostomy. Decannulation was achieved in 12 patients (median age, 42 months), and noninvasive positive pressure ventilation (NPPV) was applied in six patients. Conclusions: Only inhomogeneous series of pediatric bilateral laryngeal palsies have been published so far, making it difficult to identify prognostic and predictive markers, as well as therapeutic guidelines. Focusing on CIBP, our data suggest that the management strategy must take into account the possibility of a delayed spontaneous recovery. NPPV is an interesting therapeutic tool in this situation. A systematic somatic assessment is mandatory to look for associated conditions. Key Words: Bilateral vocal fold paralysis, congenital, idiopathic, tracheotomy, noninvasive positive pressure ventilation. Level of Evidence: 4 Laryngoscope, 00:000–000, 2014
INTRODUCTION Bilateral vocal fold paralysis is a cause of stridor and respiratory distress in neonates and infants. Early diagnosis can be made by fiberoptic examination of the larynx, but a significant proportion of the children will need immediate intubation after birth because of intense respiratory distress. Bilateral paralysis represents from 30% to 60% of all laryngeal anomalies in children.1–5 Its etiologies may be traumatic (delivery, surgery), neurological, and idiopathic, with an estimated incidence of 40%, 40%, and 30%, respectively.6 Traumatic etiology of a bilateral laryngeal palsy is commonly an easy diagnosis, and a neurologically associated condition can usually be
From the Department of Pediatric Otolaryngology–Head and Neck Surgery, Necker Enfants-Malades Hospital, Paris, France Editor’s Note: This Manuscript was accepted for publication November 10, 2014. Presented at the 2013 meeting of the French Otolaryngology Society (SFORL), Paris, France, October 2013, and the SIFORL Meeting, Montreal, Canada, June 2014. The authors have no funding, financial relationships, or conflicts of interest to disclose. Send correspondence to Nicolas Leboulanger, MD, Department of Otolaryngology–Head and Neck Surgery, Necker Enfants Malades Hospital, 149 rue de Se`vres, 75015 Paris, France. E-mail: nicolas.leboulanger@ nck.aphp.fr DOI: 10.1002/lary.25076
Laryngoscope 00: Month 2014
identified by additional explorations. When no specific cause is found, the condition is considered idiopathic. Management options in congenital bilateral laryngeal paralysis extend from watchful waiting to early tracheostomy. Several therapeutic options are possible including numerous surgical procedures. The possibility of a spontaneous recovery must always be kept in mind. Most of the reports published in the international literature so far address both unilateral and bilateral paralysis and encompass several different etiologies. To our knowledge, no series of patients has specifically focused on the evolution, management, and outcomes of congenital idiopathic bilateral laryngeal paralysis (CIBP). Therefore, the aim of this study was to report a series of pediatric patients presenting only with CIBP to better define the prognosis of this specific condition and analyze its functional outcome with regard to management strategy, decannulation rates for children who required a tracheostomy, restoration of the airway, dysphonia, and swallowing function.
MATERIALS AND METHODS We performed a retrospective review over a 15-year period (1998–2013) of all children treated in our institution for CIBP. In all patients, the diagnosis was eventually made by fiberoptic examination of the larynx. An endoscopic examination
Lesnik et al.: Idiopathic Bilateral Laryngeal Palsy
1
TABLE I. Patients. Patient
Sex
Age at Diagnosis, d
Initial Symptoms
Preterm?
Associated Condition
Intubation at Birth?
Duration, d
1
F
3
2 3
F F
60 120
4
F
7
Respiratory distress
No
Severe GERD
Yes
60
5 6
F F
21 0
Respiratory distress Stridor 1 dysphonia
No No
Severe GERD
Yes No
21
7
M
1,100
Respiratory distress
Yes
HMD
Yes
60
8 9
F M
480 1,825
Respiratory distress Respiratory distress
Yes Yes
BPD, PDA HMD, BPD, autism
Yes Yes
90 120
10
F
3
Respiratory distress
No
Yes
30
11 12
F M
21 2
Respiratory distress Stridor
No No
21
Esophageal atresia
Yes No
13
M
60
Respiratory distress
No
RDS
Yes
60
14 15
M M
15 0
Dyspnea at exertion Stridor
No No
16
F
0
Stridor
Yes
17 18
F F
0 8
Stridor Stridor
Yes Yes
19
F
60
Respiratory distress
No
Yes
60
20 21
M F
7 0
Respiratory distress Respiratory distress
No No
Yes Yes
60 21
22
M
90
Respiratory distress
Yes
Yes
180
23 24
F F
0 45
Respiratory distress Respiratory distress
No No
Yes Yes
180 10
25
M
120
Respiratory distress
Yes
Yes
10
26
M
540
Respiratory distress
Yes
Yes
?
Stridor
No
No
—
Stridor Respiratory distress
No No
No Yes
— 8
No Yes
3
HMD
Yes
90
HMD and BPD
No No
HMD
HMD
BPD 5 bronchopulmonary dysplasia; F 5 female; GERD 5 gastroesophageal reflux disease; HMD 5 hyaline membrane disease; M 5 male; PDA 5 patent ductus arteriosus; RDS 5 respiratory distress syndrome.
of the airway was also performed in all patients looking for associated airway malformations. To confirm the idiopathic type of the paralysis and assess its clinical impact, the following exams were systematically performed in each patient, during the first months of life for newborns and neonates, and after the diagnosis was made for the few late-diagnosed patients: neuropediatric clinical examination, cardiac ultrasonography, cerebral magnetic resonance imaging (MRI) (after 4 months of age), esophageal pH test, chest radiography, and barium swallow. All children presenting with a postoperative or an obstetrical (following dystocia) bilateral vocal fold paralysis were excluded. Our institutional ethics committee approved this study.
RESULTS During the considered period, 154 children were diagnosed with a laryngeal paralysis. Among them, 26 presented with a CIBP and were included in the study, which included 16 females and 10 males.
Patients The diagnosis of bilateral laryngeal palsy was made by fiberoptic examination of the larynx at a median age Laryngoscope 00: Month 2014
2
of 18 days in the intensive care unit or in the operating room for patients intubated at birth. The mean age at diagnosis would not be representative of the series because of some patients (patients 7, 8, 9, 26) who were diagnosed with a considerable delay. Nineteen patients (73%) required tracheal intubation at birth, for a mean duration of 60 days (range, 3– 180 days). Comorbidities were found in 11 patients, but none known to be responsible for a bilateral laryngeal paralysis. Nineteen children presented eventually with failure to thrive and required temporary feeding by nasogastric tube. No patients required a gastrostomy tube. Patients’ data are summarized in Tables I and II.
Initial Management An endoscopic assessment under local followed by general anesthesia was performed in all patients after 2 months (median) of age. Eleven patients displayed a CIBP in full adduction and 15 in subtotal adduction. No patients had a CIBP in abduction. Some patients presented minor anomalies of the airway (narrow cricoid and subglottis, n 5 4; suprastomal granuloma, n 5 7), but not sufficient enough to significantly reduce the airway. Lesnik et al.: Idiopathic Bilateral Laryngeal Palsy
TABLE II. Management. Initial Management
Secondary Management Age at Tracheotomy, d
Extubation failure?
Tracheotomy?
1
No
2 3
No No
4
Yes
Yes
5 6
Yes No
Yes No
7
Yes
No
No
BC, cricoid split
2
8 9
No No
No Yes
2,000
No No
BC, cricoid split BC, arytenoidectomy
2 4
10
Yes
Yes
33
No
BC, arytenoidectomy
4
Yes
11 12
Yes Yes
Yes Yes
21 60
No No
BC, arytenoidectomy UC
3 1
No Yes
13
Yes
Yes
50
14 15
No No
No No
16
Yes
Yes
17 18
No No
No No
19
Yes
Yes
58
No
UC, arytenoidectomy
3
Laryngoplasty
2
No
20 21
Yes Yes
Yes Yes
65 19
No No
BC, arytenoidectomy
5
Laryngoplasty
1
Yes No
22
Yes
Yes
180
No
23 24
Yes No
Yes No
190
No No
UC, arytenoidectomy BC
2 2
Arytenoidopexy
1
No
BC
3 Laryngoplasty
4
Patient
Watchful Waiting?
Endoscopic Procedure
No.
No
No
UC, arytenoidectomy
2
No
No No
No Yes
UC, arytenoidectomy
1
No No
66
No
UC, arytenoidectomy
2
15
No Yes
25
No
No
26
Yes
Yes
100
External Approach
No.
No Yes No No Laryngoplasty Laryngoplasty
1 1
No No
No
BC, arytenoidectomy
7
No
No Yes
BC
2
No No
No
UC, arytenoidectomy
1
No
Yes Yes
630
NPPV
Yes Yes
Yes
No
No No No Yes
BC 5 bilateral cordotomy; NPPV 5 noninvasive positive pressure ventilation; UC 5 unilateral cordotomy.
In all patients, the CIPB was found to be the only cause of upper airway obstruction. All patients eventually underwent the series of examinations described above, confirming the diagnosis of CIBP. All patients were also systematically treated for gastroesophageal reflux by proton pump inhibitors. Among the 19 patients intubated at birth, 14 (74%) could not be extubated and therefore underwent a tracheostomy at a median age of 62 days.
Secondary Management An endoscopic cordotomy was performed in 17 patients: unilateral in seven patients, including three with a tracheostomy; bilateral in 10 patients, including six with a tracheostomy. Mean age at cordotomy was 25 months. Among those 17 patients, 10 (nine with a tracheostomy) secondarily needed an endoscopic arytenoidectomy at a mean age of 33.8 months. None of our patients had a posterior cricoid split with cartilage graft. Fourteen patients underwent at least two endoscopic procedures, and six patients needed a complemenLaryngoscope 00: Month 2014
tary surgery by external approach. Procedures are detailed in Table II. Noninvasive positive pressure ventilation (NPPV) by continuous airway pressure at sleep was initiated in eight patients, including seven with a tracheostomy. In these patients, the NPPV aimed to facilitate the decannulation. No NPPV was initiated in a neonate or for pulmonary reasons.
RESULTS The mean follow-up duration was 6.7 years (range, 2–15 years). Spontaneous recovery occurred in 17 patients (65%), including nine with a tracheostomy, at a mean age of 25 months. Among the 14 patients with a tracheostomy, 12 (85%) were eventually weaned from the cannula, at a mean age of 36 months. Four patients still have NPPV at sleep. Results are summarized in Table III.
DISCUSSION This retrospective study reports on 26 patients presenting with CIBP with regard to the outcomes and Lesnik et al.: Idiopathic Bilateral Laryngeal Palsy
3
TABLE III. Results.
Patient Tracheotomy? NPPV
Weaning From NPPV?
Age at Spontaneous Spontaneous Recovery? Recovery, m Decannulation?
Age at Decannulation, m
Current Clinical Status
Actual Follow-up, y
Stridor at exertion
2.5
1
No
No
Non
2
No
No
Yes
10
Dysphonia
3
3 4
No Yes
No No
Yes Yes
12 12
Intermittent stridor Yes
31
3.25 5
5
Yes
Yes
Yes
15
Yes
30
6 7
No No
No No
Yes Yes
8 48
Excellent
7 10
21
4.5 years old
8
8
No
No
Yes
9 10
Yes Yes
No Yes
No No
11
Yes
No
Yes
12
No
12 13
Yes Yes
Yes No
Yes Yes
20 84
Yes Yes
14
No
No
Yes
15 16
No Yes
No No
Yes No
17
No
Yes
No
NPPV
1
18 19
No Yes
Yes No
Yes No
NPPV
7 4
20
Yes
Yes
21 22
Yes Yes
No Yes
23
Yes
No
No
24 25
No No
No No
Yes No
26
Yes
Yes
No
8 years old
3.6 No Yes
60 11 26
Tracheotomy NPPV
10 5
Cannula capped all day
6.5
Decannulated, NPPC
2.5 8
Stridor at exertion
16
24 No
5 5.5
54
Yes
42
Yes
72
Yes
72
NPPV
8
Yes Yes
3 21
Yes Yes
8 23
Excellent NPPV
1 8
Yes
192
Dysphonia
17
Excellent
2.3 15
NPPV
10
14 Yes
84
NPPV 5 noninvasive positive pressure ventilation.
decannulation rates. The overall results were acceptable for children who needed a tracheostomy, with a decannulation rate of 85%. CIBP is an uncommon condition in pediatric otolaryngology, and its exact incidence is not known but is probably
