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PubMed1997

[Treatment of children with congenital diseases of the larynx and trachea].

+33 cases of vascular tumors, 15 of congenital membranes, 12 of papillomatosis, 5 of cysts, 12 of abnormal laryngeal cartilages, 3 of tracheomalacia, 9 of stridor, 1 of fibrous polyp and 1 of mucous membrane folding in the interarytenoid region are reported. Operative interventions primarily endoscopic laser surgery were performed in children with congenital vascular tumors, membranes, cysts. Removal of papillomas in congenital papillomatosis conducted with the use of micro-instruments and CO2-laser was combined with immunomodulation. Children with laryngeal cartilage anomalies, tracheomalacia and stridor were subjected to prolonged nasotracheal intubation of even tracheotomy only in case of severe respiratory failure..

Child↗
PubMed1997

Surgical management of extrathoracic tracheal collapse in two large-breed dogs.

Collapse of the cervical portion of the trachea was diagnosed for 2 young large-breed dogs. Clinical signs included worsening respiratory stridor and coughing exacerbated by exercise. The diagnosis was confirmed by use of conventional radiography and fluoroscopy of the trachea. A polypropylene, spiral ring, extraluminal, tracheal prosthesis was used to successfully treat tracheal collapse in each dog. Although tracheal collapse typically affects middle-aged and old small-breed dogs, tracheal collapse should be considered as a differential diagnosis for large-breed dogs with a honking cough and respiratory stridor.

Animals↗
PubMed1997

Recessive epidermolysis bullosa simplex associated with plectin mutations: infantile respiratory complications in two unrelated cases.

Plectin is a 500 kDa protein involved in cytoskeleton-plasma membrane attachment with a wide tissue distribution including cutaneous and airway epithelia, muscle and neuronal tissue. Recently, mutations in the gene encoding plectin (PLEC1) have been implicated in the pathogenesis of an autosomal recessive variant of epidermolysis bullosa simplex in which cutaneous blistering starting in the neonatal period is associated with muscular dystrophy in later life. In this study, we report two unrelated patients, both of consanguineous parentage, who presented with cutaneous blistering and a hoarse cry from birth. Both experienced inspiratory stridor and respiratory distress, necessitating emergency tracheostomy in one case. Immunoreactivity to monoclonal antibodies against plectin was absent or markedly reduced in skin biopsies from both patients. Electron microscopy revealed a low intraepidermal plane of cleavage and hypoplastic hemidesmosomes with a reduced association with keratin intermediate filaments. Direct sequencing of PLEC1 in each case demonstrated two novel homozygous frameshift deletion mutations, 5069del19 and 5905del2, which both create downstream premature termination codons. Although currently neither patient has symptoms of muscle disease, the identification of mutations in PLEC1 may be predictive for the future development of muscular dystrophy. Recessive epidermolysis bullosa simplex resulting from abnormalities in plectin should be considered in the differential diagnosis blistering, hoarseness and stridor in infancy.

Child, Preschool↗
PubMed1997

[Compromized postadenoidectomy respiration in a child with obstructive sleep apnea syndrome].

A two-year-old boy with a history of slow growth, snoring during sleep and adenoid hypertrophy underwent adenoidectomy and transtympanic drainage under general anesthesia. Immediately after extubation, severe inspiratory stridor and shallow labored breathing began and persisted over a period of two hours, in spite of corticoid administration and oxygen therapy. The signs receded partially when the patient was seated and with a mandibular traction maneuver. As symptoms persisted, foreign body obstruction was ruled out by examination of the cavum and upper airway under general anesthesia and with orotracheal intubation. The patient was transferred to the pediatric intensive care unit, where he remained intubated for 18 hours. After extubation, stridor and shallow labored breathing reappeared but gradually receded as the residual effects of sedation disappeared. The parents mentioned symptoms suggestive of obstructive sleep apnea syndrome (OSAS) occurring since the boy was 6 months old and that had worsened in recent months. OSAS in children is characterized by intermittent obstruction of the upper airway during sleep, causing snoring and periods of apnea/hypopnea that lead to hypoxemia and hypercapnia. The most frequent cause is hypertrophy of the adenoid and tonsils, and the treatment of choice is adenotonsillectomy, although the risk of postoperative respiratory distress in such children is high. It is important to rule out OSAS in children who are candidates for adenotonsillectomy so that such patients are not scheduled for ambulatory surgery, but rather given adequate postoperative monitoring and treatment.

Adenoidectomy↗
PubMed1984

Respiratory failure in myasthenia gravis due to vocal cord paresis.

Paroxysmal dyspnea and stridor in a patient with myasthenia were shown to be due to weakness of vocal cord abductors that improved with anticholinesterase therapy. Despite adequate inspiratory force, breathing was severely impaired by increased inspiratory resistance. The inspiratory flow-volume loop was useful in documenting the laryngeal obstruction and monitoring the effect of therapy. Reinterpretation of previous studies suggests that upper airway obstruction may often contribute to respiratory failure in patients with myasthenia.

Airway Obstruction↗
PubMed1999

Congenital airway abnormalities in patients requiring hospitalization.

OBJECTIVE: To determine the cause of congenital airway abnormalities in pediatric patients requiring hospitalization for their respiratory status. DESIGN AND SETTING: Case series in a tertiary care center. PATIENTS: A 5-year retrospective chart review was conducted at our institution. A total of 174 patients were identified who required hospitalization for their respiratory status as a result of a congenital airway abnormality. RESULTS: Of the 174 patients, 114 (65.5%) were male and 60 (34.5%) were female. Eighty patients (47%) presented within the first 3 months of life. Forty-six patients (26%) were born prematurely, and 49 patients (28%) were diagnosed as having gastroesophageal reflux. The majority of patients (139 [80%]) had multiple presenting symptoms or signs. Stridor was the most common (129 [74%]), followed by accessory respiratory effort, cyanosis, apnea, and failure to thrive. Diagnosis was made at the time of surgical evaluation in 91% of the patients, with the remaining diagnoses made using radiological findings and/or clinical evaluation. Sixty-five patients (37%) had multiple sites of airway abnormalities; laryngeal abnormalities were noted almost 3 times as often as tracheal abnormalities (161 vs 62, respectively). Of the laryngeal abnormalities, laryngomalacia was the most common, followed by glottic web, subglottic stenosis, vocal-cord paralysis, and subglottic hemangioma. Tracheomalacia was the most common tracheal abnormality, followed by external compression and tracheal stenosis. Thirty-three patients (19%) required tracheotomy for management of recurrent respiratory decompensation. CONCLUSIONS: While congenital airway abnormalities are usually self-limited, those patients requiring hospitalization represent a group with a more severe respiratory status who have a greater chance of requiring tracheotomy. The recognizable percentage of patients with gastroesophageal reflux and prematurity accounts for comorbid factors in the need for hospitalization for respiratory issues related to congenital airway abnormalities.

Age Distribution↗
PubMed2000

Relapsing polychondritis: a course over 20 years with cerebral involvement.

Relapsing polychondritis is a chronic inflammatory disease associated with an autoimmune disorder in cartilaginous tissue, eyes, labyrinth, blood vessels, and central nervous system. We describe a 75-year-old woman who presented with a 20-year history of dyspnea, inspiratory stridor, and polyarthritis. She developed dysmorphism of both ears and a saddle nose approximately 10 years earlier. Subsequently, she suffered from hearing loss and a tremor. A T2-weighted magnetic resonance imaging scan of the brain revealed multiple, spotted signal intensities. Immunohistochemical analysis of a serum sample showed antibodies to cartilaginous tissue, which were further identified on immunoblotting as antibodies to type II collagen. The extremely prolonged course of disease (>20 years) until a correct diagnosis was made is remarkable. Also, cerebral involvement, which was most likely caused by cerebral angiitis, and which, to our knowledge, has never previously been reported in this form, was detected. Arch Otolaryngol Head Neck Surg. 2000;126:1495-1498

Aged↗
PubMed2001

Graft-vs-host disease as a cause of enlargement of the epiglottis in an immunocompromised child.

We report a rare case of dyspnea due to enlargement of the epiglottis in a severely immunocompromised patient. The child underwent a previous tracheostomy at another hospital because of respiratory distress under the diagnosis of acute epiglottitis. The patient was subsequently decannulated without incident. One year later, the child developed a new episode of dyspnea with inspiratory stridor. A new tracheostomy was neccessary, and a biopsy specimen of the enlarged epiglottis was taken to confirm the diagnosis of graft-vs-host disease. The therapeutic measures in these situations are discussed below, and a review of the current literature concerning the etiology and management of epiglottic enlargement is performed.

Child, Preschool↗
PubMed2001

Use of rigid and flexible bronchoscopy among pediatric otolaryngologists.

OBJECTIVE: To explore how rigid and flexible bronchoscopy are used in pediatric otolaryngologic practice. DESIGN: Survey. PARTICIPANTS: Members of the American Society of Pediatric Otolaryngology who practice in the United States and Canada and were listed in the membership directory were eligible. Of the 206 members, 24 practicing outside the United States or Canada and 11 without an e-mail address or a fax machine were excluded. Hence, a questionnaire was e-mailed or faxed to 171 pediatric otolaryngologists. MAIN OUTCOME MEASURES: Questions concerned the practice setting, type and number of bronchoscopies, indications, complications, and medicolegal cases. RESULTS: Responses were received from 120 subjects (70.2%), with 3 retired and 2 practicing only otology, leaving 115 respondents who completed at least some of the questionnaire. Rigid and flexible bronchoscopy were performed by 72.7% (56/77) of those in academic settings and by 71.1% (27/38) of those in group or solo practices. In the last 12 months, approximately 10 454 total bronchoscopies were performed, with 2052 flexible and 9117 rigid bronchoscopies. Stridor, suspected foreign body inhalation, and laryngomalacia were the most common indications for bronchoscopy. Of the 83 respondents practicing rigid and flexible bronchoscopy, 25 (30.1%) used both instruments to manage complex or repeated foreign bodies, 25 (30.1%) used both to manage patients with cystic fibrosis, and 15 (18.1%) used both to manage simple foreign bodies. Complications were reported by 15.7% of the respondents, the most common being arrhythmia. Familiarity with a case resulting in medicolegal action was reported by 32.2% of the respondents. CONCLUSIONS: Rigid and flexible bronchoscopy have multiple uses in pediatric otolaryngologic practice. Also, flexible bronchoscopy appears to be emerging as a more frequently used diagnostic and therapeutic tool.

Bronchoscopes↗
PubMed2001

Localization of the gene for familial laryngeal abductor paralysis to chromosome 6q16.

BACKGROUND: Vocal fold paralysis is a common cause of neonatal stridor. Although it is usually classified as idiopathic or iatrogenic in origin, a small subset of patients have a family history of this disorder, indicating a possible genetic cause. OBJECTIVE: To identify the genetic locus of the gene that causes familial laryngeal abductor paralysis. DESIGN: A standard nonorganic protocol was used to extract DNA from whole-blood samples. The DNA samples were quantified by DNA fluorometry, and the concentration of all samples was standardized at 40 ng/microL. A pooled DNA strategy was used to facilitate rapid polymerase chain reaction screening of markers in the Weber v8.0 genome screening set. Polymerase chain reaction screening of individual DNA samples was performed using possible linked markers initially identified as having an allele that appeared with a higher incidence in the affected DNA pools. Statistical analysis of possible linkage was performed using the LINKAGE 5.1 set of linkage analysis computer programs. SUBJECTS: A family in which a form of familial laryngeal abductor paralysis segregates was ascertained. Whole blood samples were drawn from 40 participating individuals within this family after the subjects' fully informed consent was obtained. RESULTS: Initial screening of the pooled DNA specimens revealed a band pattern for D6S1021 on chromosome 6q16, indicating an allele with a higher incidence in the affected vs the nonaffected pool. Two-point analysis of individual allele patterns confirmed linkage to D6S1021 with an lod score of 3.86 (straight theta = 0.0) at a penetrance value of 0.8. Haplotype analysis with flanking markers defined a 5-centiray critical region between D6S283 and AFMA047YG1. CONCLUSION: An autosomal dominant form of familial laryngeal abductor paralysis is linked to a 5-centiray region on chromosome 6q16 surrounding D6S1021.

Alleles↗
PubMed2002

Indications for tracheotomy in the pediatric intensive care unit population: a pilot study.

OBJECTIVE: To define the indications for tracheotomy in patients requiring prolonged intubation (>1 week) in the pediatric intensive care unit (PICU). DESIGN: Retrospective chart review and follow-up telephone survey. SETTING: A tertiary care center PICU. OUTCOME MEASURE: Tracheotomy or extubation. PATIENTS: All patients older than 30 days in the PICU intubated for longer than 1 week between 1997 and 1999. RESULTS: During the study, 63 total admissions required intubation for longer than 1 week. A tracheotomy was necessary in 14% of admissions (n = 9). The mean length of intubation before the tracheotomy was 424 hours, whereas the mean length of intubation without the need for tracheotomy was 386 hours. Length of intubation, age, and number of intubations did not increase the probability of having a tracheotomy. Of those requiring a tracheotomy, 2 had tracheomalacia, 1 had subglottic edema, 1 had plastic bronchitis, 1 had Down syndrome with apnea resulting in right heart failure, 3 required long-term ventilation after cardiopulmonary collapse, and 1 had mitochondrial cytopathy. Of these 9 children, 7 were successfully decannulated, 1 patient died of underlying disease, and 1 patient remained cannulated secondary to the mitochondrial cytopathy. Twenty families of the patients who did not undergo a tracheotomy were reached by telephone after discharge. Most of the families reported that their children were free of stridor and hoarseness after extubation. CONCLUSIONS: Children tolerate prolonged intubation without laryngeal complications. The consideration for tracheotomy in the PICU setting must be highly individualized for each child.

Airway Obstruction↗
PubMed2002

Laryngeal duplication cyst.

Benign congenital laryngeal cysts are rare. Infants and children with these lesions can present with chronic or intermittent airway obstructive symptoms, hoarseness, aspiration, chronic cough, or failure to thrive. The most common congenital laryngeal cysts include saccular cysts, laryngoceles, and ductal cysts. Other more unusual laryngeal lesions, such as hamartomas, choristomas, and teratomas, can also present with these symptoms. We describe a unique congenital cyst that arose in continuity with the larynx in a child with hoarseness and intermittent stridor. The features of this lesion are similar to those of bronchogenic duplication cysts of the trachea, but with histopathological features of the larynx.

Abnormalities, Multiple↗
PubMed2002

Anesthetic complications of tympanostomy tube placement in children.

OBJECTIVE: To determine the incidence of perioperative anesthesia complications during bilateral myringotomy with tympanostomy tube placement (BMTT). SETTING: Tertiary care children's hospital where otolaryngology attending physicians and residents performed surgical procedures. Anesthesia providers included pediatric anesthesiologists, residents, nurse anesthetists, and students. METHODS: Medical record review was performed for a consecutive series of 3198 children undergoing BMTT (1000 prospectively, 2198 retrospectively). For the prospectively studied patients, major adverse events, which included laryngospasm and stridor, and minor adverse events, including upper airway obstruction, prolonged recovery, emesis, and persistent postprocedural agitation, were noted. Also recorded were the patient's American Society of Anesthesiologists (ASA) physical class status, age, concurrent medical conditions, and type of anesthesia provider. RESULTS: Fewer than 9% of prospectively studied pediatric patients experienced a minor adverse event, whereas a major event occurred in 1.9%. Eighty-one percent of the events experienced were attributable to agitation or prolonged recovery. Neither ASA status (P =.38), age (P =.15), nor type of anesthesia provider (P =.06) were significantly related to the occurrence of an adverse event. However, a child with an acute or chronic illness has 2.78 times the odds of experiencing an adverse event compared with a child with no illness (P<.001). CONCLUSIONS: Anesthesia administered for placement of tympanostomy tubes by physicians who specialize in the care of children in a tertiary care children's hospital is safe. The most significant predictor of a minor anesthetic event during BMTT is the presence of a preexisting medical condition or concurrent acute illness.

Acute Disease↗
PubMed2003

Extraesophageal reflux in pediatric patients with upper respiratory symptoms.

OBJECTIVE: To systematically review published literature describing the association between reflux and upper airway symptoms in children. DESIGN: Structured MEDLINE search of English-language articles published since 1966. SUBJECTS: We selected articles examining reflux in conjunction with stridor, apnea, sudden infant death syndrome, life-threatening events, and laryngomalacia. Studies that focused on lower airway symptoms or adults were excluded. OUTCOME MEASURES: Articles were abstracted for patient factors, elements of study design, methods of reflux diagnosis, and definition of pathologic reflux. RESULTS: Ninety-nine articles were identified, 56 of which specifically examined reflux and upper respiratory symptoms in children. Of these, 10 compared reflux incidence in symptomatic patients and a set of predetermined control patients, while the remainder reported prevalence data only. Overall, symptomatic patients were diagnosed with reflux frequently, with a range from 27% to 100%. In studies that attempted to compare patients with controls, only 2 provided statistical comparisons of the patient groups, and none adjusted for confounding owing to study design. There was marked heterogeneity in methods used to diagnose reflux in the studies reviewed, with only 34% using dual-channel pH testing; definitions of pathologic reflux were also variable. CONCLUSIONS: Evidence seems to support the hypothesis that reflux is associated with upper airway symptoms in children. However, the strength of this correlation and the risk of upper airway symptoms attributable to reflux are difficult to determine given the limitations of available literature. Future research studies should seek standard reflux testing methods, clear comparison groups, and more rigorous statistical methods.

Causality↗
PubMed2004

The biology and management of laryngeal neurofibroma.

OBJECTIVES: To review the presentation of laryngeal neurofibroma, including its association with neurofibromatosis types 1 and 2, and present guidelines for its management. DESIGN: Retrospective study. PATIENTS: Five pediatric patients with laryngeal neurofibroma, 4 girls (80%) and 1 boy (20%), were treated at a tertiary pediatric medical center from 1973 through 2003. Recorded data included age at initial presentation, sex, symptoms, significant medical and family history, preoperative evaluation, location of the tumor, surgical procedure, complications, outcome, and recurrence. RESULTS: The 5 patients presented with stridor and cafe-au-lait spots at or shortly after birth. All patients were diagnosed as having neurofibromatosis type 1 by the established criteria. Studies evaluating the disease processes included plain radiography, computerized tomography, magnetic resonance imaging, barium swallow, and laryngoscopy and bronchoscopy under anesthesia. Pathologic examination of biopsy specimens from all patients showed neurofibromas with plexiform and/or diffuse features. Treatments included tracheotomy (n = 4), carbon dioxide laser excision (n = 4), modified neck dissection (n = 3), partial pharyngectomy (n = 1), supraglottic laryngectomy (n = 1), and endoscopic hemilaryngectomy (n = 1). Three patients were successfully decannulated. Follow-up ranged from 1 to 15 years. One patient was lost to follow-up. No evidence of malignant degeneration was noted. CONCLUSIONS: Neurofibroma of the larynx is a rare condition that should be considered in the differential diagnosis of children with a submucosal laryngeal mass. In our series, all patients had associated neurofibromatosis type 1. Complete surgical excision is the treatment of choice in cases of localized small lesions. To prevent debilitating outcomes due to aggressive surgery, minimally invasive procedures (partial excision via endoscopic approach) may be preferable for larger lesions that infiltrate the surrounding vital structures. Long-term follow up of these patients is essential owing to the possibility of malignant transformation.

Adolescent↗
PubMed2004

Age-, site-, and time-specific differences in pediatric deep neck abscesses.

OBJECTIVE: To clarify presentations, organisms, and locations of deep neck abscesses in children. DESIGN: Retrospective review. SETTING: Tertiary children's hospital. The study population comprised 169 patients younger than 19 years who were surgically treated for deep neck abscesses between 1989 and 1999. MAIN OUTCOME MEASURE: Resolution of abscess. RESULTS: Neck mass (91%), fever (86%), cervical adenopathy (83%), poor oral intake (66%), and neck stiffness (59%) were common in all ages. Patients younger than 4 years, compared with patients 4 years or older, presented with agitation (50% vs 14%), cough (35% vs 14%), drooling (27% vs 12%), lethargy (46% vs 33%), oropharyngeal abnormalities (45% vs 60%), respiratory distress (5% vs 2%), retractions (5% vs 2%), rhinorrhea (53% vs 15%), stridor (4% vs 2%), and trismus (14% vs 53%). Children younger than 1 year were infected with Staphylococcus aureus (79%) vs group A streptococcus (6%). Children 1 year or older were infected with group A streptococcus (29%) vs S aureus (16%). Retropharyngeal or parapharyngeal regions were involved in children 1 year or older (49%) vs younger than 1 year (21%). Anterior or posterior triangles and submandibular or submental regions were involved in 39% and 36%, respectively, of children younger than 1 year vs 30% and 23%, respectively, of children 1 year or older. Retropharyngeal and parapharyngeal abscesses yielded group A streptococcus (34%) vs S aureus (11%). Anterior and posterior triangle abscesses yielded S aureus (35%) vs group A streptococcus (19%), as did submandibular and submental abscesses (42% vs 19%). CONCLUSIONS: Abscesses in children younger than 1 year affected anterior or posterior triangles and submandibular or submental regions, yielding S aureus. Abscesses in children 1 year or older affected retropharyngeal or parapharyngeal regions, yielding group A streptococcus.

Abscess↗
PubMed2004

The use of posterior cricoid grafting in managing isolated posterior glottic stenosis in children.

OBJECTIVE: To review our experience using posterior cricoid grafts to manage pediatric patients with isolated posterior glottic stenosis (PGS). DESIGN: Retrospective review over a 12-year period. SETTING: Tertiary care pediatric hospital. PATIENTS: All patients with isolated PGS treated between 1990 and 2002, in whom PGS was the dominant airway lesion and laryngotracheoplasty was required. Patients with concomitant vocal cord paralysis, a history of posterior laryngeal clefting, a Bogdasarian type I stenosis, or subglottic stenosis worse than grade I were excluded. MAIN OUTCOME MEASURES: Cause, operative intervention, decannulation rate, failure rate, and requirement for secondary procedures. RESULTS: A total of 29 patients ranging in age from 2 to 8 years were treated (21 with a history of prolonged intubation and 8 with a history of laryngeal trauma). Twenty patients had tracheotomies in place at the time of airway reconstruction and the remainder had stridor. Costal cartilage was the preferred graft material and was used in 27 patients. Six patients were referred with a diagnosis of bilateral vocal cord paralysis, but on evaluation were found to have PGS and mobile vocal cords. In 12 patients, repair was accomplished in a single-stage procedure; a suprastomal stent was placed in 17 patients. Overall decannulation rate was 97%, though a second procedure was required in 4 patients. One patient remained tracheotomy dependent; 4 had poor voice, including 2 with a history of laryngeal fracture; and 2 had late arytenoid prolapse. CONCLUSIONS: Isolated PGS in children is effectively managed with costal cartilage grafting of the posterior cricoid. This series has seen an evolution in management, with shorter stenting periods, placement of flanged posterior grafts without sutures, and graft placement without complete laryngofissure.

Adolescent↗
PubMed2004

Tissue-engineered cartilage as a graft source for laryngotracheal reconstruction: a pig model.

OBJECTIVE: To evaluate the feasibility of using tissue-engineered cartilage for laryngotracheal reconstruction in the pig model. DESIGN: Auricular cartilage was harvested from 3 young swine. The cartilage was digested, processed, and suspended and a cell culture was obtained. The cells were then suspended in 3 mL of a 30% solution of a biodegradable polymer (Pluronic F-127) (polyethylene oxide/polypropylene oxide copolymer) at a cellular concentration of 50 x 10(6) cells/mL. This suspension was then implanted subcutaneously into each pig's dorsum. Eight weeks after implantation, the cartilage was harvested with the surrounding perichondrial capsule. An anterior cartilage graft laryngotracheal reconstruction was performed. Bronchoscopy was performed at 3 postoperative weeks to demonstrate airway patency. The animals were killed at 3 months, and specimens were obtained for histological analysis. SETTING: An animal research facility. SUBJECTS: Three young Yorkshire swine. RESULTS: All 3 pigs survived to the 3-month postoperative interval with no evidence of stridor or airway distress. Interval bronchoscopy revealed a normal patent airway with a mucosalized graft. Histopathologic analysis revealed incorporation of the tissue-engineered cartilage graft in the cricoid area, which correlated with results of bronchoscopic evaluation. CONCLUSION: Tissue-engineered auricular cartilage served as a viable graft in the pig model and might be an alternative cartilage source for laryngotracheal reconstruction.

Animals↗