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1.
Plast Reconstr Surg ; 144(3): 704-709, 2019 09.
Artigo em Inglês | MEDLINE | ID: mdl-31461034

RESUMO

BACKGROUND: Apert syndrome is frequently combined with respiratory insufficiency, because of the midfacial deformity which, in turn, is influenced by the malformation of the skull base. Respiratory impairment resulting from Apert syndrome is caused by multilevel limitations in airway space. Therefore, this study evaluated the segmented nasopharyngeal and laryngopharyngeal anatomy to clarify subcranial anatomy in children with Apert syndrome and its relevance to clinical management. METHODS: Twenty-seven patients (Apert syndrome, n = 10; control, n = 17) were included. All of the computed tomographic scans were obtained from the patients preoperatively, and no patient had confounding disease comorbidity. Computed tomographic scans were analyzed using Surgicase CMF. Craniometric data relating to the midface, airway, and subcranial structures were collected. Statistical significance was determined using t test analysis. RESULTS: Although all of the nasal measurements were consistent with those of the controls, the nasion-to-posterior nasal spine, sphenethmoid-to-posterior nasal spine, sella-to-posterior nasal spine, and basion-to-posterior nasal spine distances were decreased 20 (p < 0.001), 23 (p = 0.001), 29 (p < 0.001), and 22 percent (p < 0.001), respectively. The distance between bilateral gonions and condylions was decreased 17 (p = 0.017) and 18 percent (p = 0.004), respectively. The pharyngeal airway volume was reduced by 40 percent (p = 0.01). CONCLUSION: The airway compromise seen in patients with Apert syndrome is attributable more to the pharyngeal region than to the nasal cavity, with a gradually worsening trend from the anterior to the posterior airway, resulting in a significantly reduced volume in the hypopharynx.


Assuntos
Acrocefalossindactilia/patologia , Laringe/anormalidades , Nasofaringe/anormalidades , Criança , Pré-Escolar , Feminino , Humanos , Lactente , Masculino , Estudos Retrospectivos
2.
Medicine (Baltimore) ; 98(28): e16364, 2019 Jul.
Artigo em Inglês | MEDLINE | ID: mdl-31305433

RESUMO

RATIONALE: Esophageal duplication cyst (EDC) is a rare developmental aberration originated from the embryonic foregut. It may remain asymptomatic but produce local mass effect on surrounding organs if rapid enlarges. EDC may sometimes accompany with other congenital malformations. Congenital pulmonary airway malformation (CPAM) is a congenital lung malformation with an unknown chance of developing symptoms. Here we report a rare case of esophageal duplication cyst with type 2 congenital pulmonary airway malformation (CPAM). PATIENT CONCERNS: A 16-month old boy with a prenatal diagnosis of type 2 CPAM presented progressive stridor and respiratory distress and was admitted to our hospital under the diagnosis of pneumonia. The patient responded poorly to antibiotics. A chest Xray (CXR) showed consolidation over the left upper lobe with trachea deviated to right side. A chest computed tomography (CT) revealed a cystic lesion sized 3.3 × 3.3 cm in the superior mediastinum. DIAGNOSES: Post-operative pathological report confirmed the diagnosis of esophageal duplication cyst. INTERVENTIONS: We pre-medicated the patient with steroids and inhaled bronchodilators for airway maintenance. Then the patient received tumor resection via median sternotomy. OUTCOMES: The patient recovered without complication and discharged smoothly 4 days after the surgery. LESSONS: EDC is a rare but potentially life-threatening disease owning to compression of large airways. Chest CT scan could detect the lesion non-invasively and should be considered in patients with persistent stridor, as well as CXR findings of the trachea deviated by a mass lesion in mediastinum, especially for those with CPAM.


Assuntos
Anormalidades Congênitas , Cisto Esofágico/complicações , Esôfago/anormalidades , Laringe/anormalidades , Pulmão/anormalidades , Sons Respiratórios , Anormalidades Congênitas/diagnóstico , Anormalidades Congênitas/terapia , Diagnóstico Diferencial , Cisto Esofágico/diagnóstico , Cisto Esofágico/terapia , Esôfago/diagnóstico por imagem , Esôfago/cirurgia , Humanos , Lactente , Pulmão/diagnóstico por imagem , Masculino , Sons Respiratórios/diagnóstico
3.
Lakartidningen ; 1162019 May 15.
Artigo em Sueco | MEDLINE | ID: mdl-31192393

RESUMO

The main, but not sole, indication for an Ex-utero Intrapartum Treatment (EXIT) delivery is an airway obstruction due to either laryngeal atresia or tumors in the head and neck region. Here we present our Institution's experience with eleven cases: three teratomas, four lymphatic malformations, two laryngeal atresias and two dermoid cysts. The EXIT procedure was used to secure the fetal airway while maintaining uteroplacental gas exchange and fetal hemodynamic stability through the umbilical circulation. Five fetuses required tracheostomy. Only one fetal death occurred due to extensive growth of a teratoma preventing us from establishing an airway. No other fetal or major maternal complication occurred. The EXIT procedure is a complex procedure and these rare cases should be referred to a center with a dedicated and experienced multidisciplinary team.


Assuntos
Obstrução das Vias Respiratórias , Histerotomia/métodos , Laparotomia/métodos , Adulto , Obstrução das Vias Respiratórias/congênito , Obstrução das Vias Respiratórias/cirurgia , Cesárea , Malformação Adenomatoide Cística Congênita do Pulmão/cirurgia , Feminino , Neoplasias de Cabeça e Pescoço/congênito , Neoplasias de Cabeça e Pescoço/cirurgia , Hospitais Universitários , Humanos , Recém-Nascido , Intubação Intratraqueal/métodos , Doenças da Laringe/congênito , Doenças da Laringe/cirurgia , Laringe/anormalidades , Laringe/cirurgia , Anormalidades Linfáticas/cirurgia , Equipe de Assistência ao Paciente , Gravidez , Resultado da Gravidez , Diagnóstico Pré-Natal , Estudos Retrospectivos , Região Sacrococcígea/patologia , Região Sacrococcígea/cirurgia , Suécia , Teratoma/congênito , Teratoma/cirurgia , Traqueotomia/métodos
4.
Int J Pediatr Otorhinolaryngol ; 121: 164-172, 2019 Jun.
Artigo em Inglês | MEDLINE | ID: mdl-30917301

RESUMO

INTRODUCTION: Congenital head and neck pathology may cause direct postnatal airway obstruction. Prenatal diagnosis facilitates safe delivery with pre- and perinatal airway assessment and management and Ex-Utero-Intrapartum-Treatment (EXIT) if necessary. Fetoscopic airway evaluation can optimize the selection of patients in need of an EXIT procedure. METHODS: Description of 11 consecutive fetuses, born with a potential airway obstruction between 1999 and 2011 and treated at the University Hospitals Leuven, with a long-term follow-up until 2018. An algorithm including fetoscopic airway evaluation is presented. RESULTS: In utero imaging revealed seven teratomas, one fourth branchial pouch cyst, one thymopharyngeal duct remnant, one lymphatic malformation and one laryngeal atresia. A multidisciplinary team could avoid EXIT in eight patients by ultrasonographic (n = 2) or fetoscopic (n = 6) documentation of accessible airways. Three patients needed an EXIT-to-airway-procedure. Neonatal surgery included tracheostomy during EXIT (n = 2) and resection of teratoma (n = 7) or branchiogenic pathology (n = 3). All patients do well at long-term (minimum 54 months) follow-up. CONCLUSIONS: Combining prenatal imaging and perinatal fetoscopy, EXIT-procedure and neonatal surgery yields an optimal long-term outcome in these complex patients. Fetoscopy can dramatically reduce the number of EXIT-procedures.


Assuntos
Obstrução das Vias Respiratórias/diagnóstico por imagem , Obstrução das Vias Respiratórias/cirurgia , Anormalidades Congênitas/diagnóstico por imagem , Fetoscopia , Neoplasias de Cabeça e Pescoço/diagnóstico por imagem , Teratoma/diagnóstico por imagem , Obstrução das Vias Respiratórias/congênito , Obstrução das Vias Respiratórias/etiologia , Algoritmos , Anormalidades Congênitas/cirurgia , Feminino , Neoplasias de Cabeça e Pescoço/complicações , Neoplasias de Cabeça e Pescoço/congênito , Neoplasias de Cabeça e Pescoço/cirurgia , Humanos , Recém-Nascido , Laringe/anormalidades , Anormalidades Linfáticas/complicações , Anormalidades Linfáticas/diagnóstico por imagem , Período Periparto , Gravidez , Teratoma/complicações , Teratoma/congênito , Teratoma/cirurgia , Fatores de Tempo , Traqueostomia , Ultrassonografia Pré-Natal
5.
Pediatr Surg Int ; 35(5): 565-568, 2019 May.
Artigo em Inglês | MEDLINE | ID: mdl-30783751

RESUMO

PURPOSE: A type IV laryngotracheoesophageal cleft (LTEC) is a very rare congenital malformation. Type IV LTEC that extends to the carina have poor prognosis and are difficult to manage. We present our experience with surgical repair in such a case using extracorporeal membranous oxygenation (ECMO). METHODS: A male infant, who was diagnosed with Goldenhar syndrome, showed severe dyspnea and dysphagia. Laryngoscopy indicated the presence of LTEC. The patient was transferred to our institute for radical operation 26 days after birth. Prior to surgery, a balloon catheter was inserted in the cardiac region of stomach through the lower esophagus to block air leakage, to maintain positive pressure ventilation. We also performed observations with a rigid bronchoscope to assess extent of the cleft, and diagnosed the patient with type IV LTEC. After bronchoscopy, we could intubate the tracheal tube just above the carina. Under ECMO, repair of the cleft was performed by an anterior approach via median sternotomy. RESULTS: The patient was intubated via nasotracheal tube and paralysis was maintained for 2 weeks, using a muscle relaxant for the first 3 days. Two weeks after surgery, rigid bronchoscopy showed that the repair had been completed, and the tracheal tube was successfully extubated without tracheotomy. CONCLUSIONS: Although insertion of a balloon catheter is a very simple method, it can separate the respiratory and digestive tracts. This method allowed for positive pressure ventilation and prevented displacement of the endotracheal tube until ECMO was established. As a result, we safely performed the operation and the post-operative course was excellent.


Assuntos
Anormalidades Congênitas/cirurgia , Esôfago/anormalidades , Esôfago/cirurgia , Oxigenação por Membrana Extracorpórea , Laringe/anormalidades , Traqueia/anormalidades , Traqueia/cirurgia , Anormalidades Múltiplas/cirurgia , Humanos , Recém-Nascido , Laringoscopia , Laringe/cirurgia , Masculino , Traqueostomia , Resultado do Tratamento
6.
Laryngoscope ; 129(11): 2588-2593, 2019 Nov.
Artigo em Inglês | MEDLINE | ID: mdl-30671968

RESUMO

OBJECTIVES: The clinical significance of the interarytenoid mucosal height (IAMH) in pediatric dysphagia, ranging from normal anatomy to a laryngeal cleft, is unknown. This study seeks to evaluate a cohort of patients who underwent evaluation of their IAMH during microdirect laryngoscopy (MDL) for associations between IAMH and dysphagia as diagnosed on preoperative videofluoroscopic swallow study (VFSS). METHODS: A retrospective case series of 1,351 patients who underwent MDL between 2011 and 2016 were reviewed for intraoperative evaluation of IAMH using our interarytenoid assessment protocol. After exclusions, 182 patients were divided into three groups: 1) thickened diet: VFSS with recommendation for thickened liquids (n = 82 of 182; 45.1%), 2) normal diet: VFSS with allowance of thin liquids (n = 19 of 182; 10.4%), and 3) control: no VFSS performed (n = 81 of 182; 44.5%). RESULTS: There was no difference in IAMH between groups (P = 0.35). Power analysis was able to achieve > 80% power to detect an effect size of ≥ 0.5 (1-5 mucosal height scale). The majority of patients in each group had an IAMH above the false vocal folds (thickened diet: 57.3%, normal diet: 57.9%, control: 64.2%). There were similar percentages of patients in each group with an IAMH at or below the true vocal folds (thickened diet: 4.9%, normal diet: 5.3%, control: 6.1%). CONCLUSION: There was no significant association between IAMH and preoperative thickened liquid recommendation in this cohort. This data fails to support the hypothesis that the IAMH is an independent etiological factor for pediatric pharyngeal dysphagia. Further studies comparing IAMH with outcomes after feeding therapy and surgery may better clarify this relationship between anatomy and physiology. LEVEL OF EVIDENCE: 4. Laryngoscope, 129:2588-2593, 2019.


Assuntos
Cartilagem Cricoide/patologia , Transtornos de Deglutição/etiologia , Transtornos de Deglutição/patologia , Mucosa Laríngea/patologia , Fotofluorografia/estatística & dados numéricos , Estatura , Criança , Pré-Escolar , Anormalidades Congênitas/etiologia , Anormalidades Congênitas/patologia , Cartilagem Cricoide/diagnóstico por imagem , Transtornos de Deglutição/diagnóstico por imagem , Feminino , Humanos , Mucosa Laríngea/diagnóstico por imagem , Laringoscopia/métodos , Laringe/anormalidades , Laringe/patologia , Masculino , Microcirurgia/métodos , Período Pré-Operatório , Estudos Retrospectivos , Gravação em Vídeo
7.
Int J Pediatr Otorhinolaryngol ; 118: 115-119, 2019 Mar.
Artigo em Inglês | MEDLINE | ID: mdl-30611096

RESUMO

OBJECTIVES: Deep interarytenoid notch (DIN) is a congenital variation of the larynx often associated with dysphagia and aspiration (DA) in young children. Feeding therapy with thickeners and surgical management with injection larygoplasty (IL) are used with various efficacies. Thickeners address the functional domain and IL addresses the anatomical domain of treatment. Our objective was to evaluate DIN patients managed with both interventions. METHODS: We conducted a retrospective pilot descriptive study of DIN patients with DA aged 1-3 years receiving thickeners and IL. Patients received a systematic weekly reduction of thickeners, referred to as the Thickener Weaning Protocol (TWP), based on clinical signs and symptoms of DA. The outcomes were assessed by the rate of thickener level reduction and DA-related sign/symptom frequency achieved at 6 months post-treatment. RESULTS: Thirteen patients with DIN associated DA were analyzed. The TWP was initiated within 2 months in 77% of patients, and within 4 months in 100% of patients. Thickener scores improved from an average of 5.76 (3/4 honey) to 2.15 (thin) (p = 0.001). DA-related signs/symptoms frequency improved from an average of 3.3 to 0.84 (p = 0.05). CONCLUSIONS: These findings suggest that treatment of DIN associated DA with a combination of thickeners and IL results in significant clinical improvements in young children.


Assuntos
Anormalidades Congênitas/terapia , Transtornos de Deglutição/terapia , Laringoplastia/métodos , Laringe/anormalidades , Aspiração Respiratória/terapia , Pré-Escolar , Transtornos de Deglutição/etiologia , Feminino , Humanos , Lactente , Injeções , Masculino , Projetos Piloto , Aspiração Respiratória/etiologia , Estudos Retrospectivos , Desmame
8.
J Voice ; 33(4): 561-563, 2019 Jul.
Artigo em Inglês | MEDLINE | ID: mdl-30224307

RESUMO

OBJECTIVES: Vocal nodules are frequent in children and tend to be reabsorbed after puberty, especially in boys. The aims of the present study were to analyze the progression of nodules in boys after puberty and to investigate the role of microweb in the persistence of these lesions. METHODS: Clinical and videolaryngoscopy evaluations were carried out in boys with vocal nodules treated between 2009 and 2016, followed up to the age of 17 years or until remission of symptoms and reabsorption of the nodules. Boys with vocal nodules who underwent regular vocal therapy are included in the study. The outcomes were remission of symptoms and of the nodular lesions. RESULTS: A total of 34 boys with vocal nodules were diagnosed, of which 21 completed follow-up. Mean age of the first evaluation was 9 years (5-11 years) and of end of treatment was 14 years (13-18 years). Mean number of videolaryngoscopy per child was five. After adolescence there was complete remission of symptoms and of the lesions in 15 children (71.4%), partial remission in four (19.0%), and no remission of symptoms and of the lesions in two cases. In these two, videolaryngoscopy identified the presence of microweb and nodules. CONCLUSIONS: Favorable reabsorption of the vocal nodules after adolescence was observed in the majority of the boys who underwent voice therapy; however, in two cases there was no remission of symptoms and of the lesions. The unfavorable course can be attributed to anterior laryngeal microwebs.


Assuntos
Anormalidades Congênitas/patologia , Doenças da Laringe/patologia , Laringe/anormalidades , Puberdade , Prega Vocal/patologia , Adolescente , Desenvolvimento do Adolescente , Fatores Etários , Criança , Desenvolvimento Infantil , Pré-Escolar , Anormalidades Congênitas/fisiopatologia , Anormalidades Congênitas/terapia , Humanos , Doenças da Laringe/fisiopatologia , Doenças da Laringe/terapia , Laringe/patologia , Laringe/fisiopatologia , Masculino , Indução de Remissão , Maturidade Sexual , Fatores de Tempo , Resultado do Tratamento , Prega Vocal/fisiopatologia , Qualidade da Voz , Treinamento da Voz
9.
Laryngoscope ; 129(7): 1696-1698, 2019 07.
Artigo em Inglês | MEDLINE | ID: mdl-30450589

RESUMO

A 12-month-old male underwent injection laryngoplasty for dysphagia associated with a deep interarytenoid notch. He subsequently developed a large postcricoid mucocele requiring revision surgery and marsupialization of the cyst just 2 weeks following the initial surgery. Laryngoscope, 129:1696-1698, 2019.


Assuntos
Anormalidades Congênitas/cirurgia , Laringe/anormalidades , Mucocele/cirurgia , Complicações Pós-Operatórias/cirurgia , Humanos , Lactente , Injeções/efeitos adversos , Laringoscopia , Laringe/cirurgia , Masculino , Reoperação
10.
Otolaryngol Head Neck Surg ; 160(3): 533-539, 2019 03.
Artigo em Inglês | MEDLINE | ID: mdl-30322357

RESUMO

OBJECTIVE: While the Benjamin-Inglis classification system is widely used to categorize laryngeal clefts, it does not clearly differentiate a type 1 cleft from normal anatomy, and there is no widely accepted or validated protocol for systematically evaluating interarytenoid mucosal height. We sought to propose the interarytenoid assessment protocol as a method to standardize the description of the interarytenoid anatomy and to test its reliability. STUDY DESIGN: Retrospective review of endoscopic videos. SETTING: Pediatric academic center. SUBJECTS AND METHODS: The interarytenoid assessment protocol comprises 4 steps for evaluation of the interarytenoid region relative to known anatomic landmarks in the supraglottis, glottis, and subglottis. Thirty consecutively selected videos of the protocol were reviewed by 4 otolaryngologists. The raters were blinded to identifying information, and the video order was randomized for each review. We assessed protocol completion times and calculated Cohen's linear-weighted κ coefficient between blinded expert raters and with the operating surgeon to evaluate interrater/intrarater reliability. RESULTS: Median age was 4.9 years (59 months; range, 1 month to 20 years). Median completion time was 144 seconds. Interrater and intrarater reliability showed substantial agreement (interrater κ = 0.71 [95% confidence interval (CI), 0.55-0.87]; intrarater mean κ = 0.70 [95% CI, 0.59-0.92/rater 1, 0.47-0.85/rater 2]; P < .001). Comparing raters to the operating surgeon demonstrated substantial agreement (mean κ = 0.62; 95% CI, 0.31-0.79/rater 1, 0.48-0.89/rater 2; P < .001). CONCLUSION: The interarytenoid assessment protocol appears reliable in describing interarytenoid anatomy. Rapid completion times and substantial interrater/intrarater reliability were demonstrated. Incorporation of this protocol may provide important steps toward improved standardization in the anatomic description of the interarytenoid region in pediatric dysphagia.


Assuntos
Cartilagem Aritenoide/patologia , Anormalidades Congênitas/diagnóstico , Laringoscopia , Laringe/anormalidades , Adolescente , Criança , Pré-Escolar , Protocolos Clínicos , Feminino , Humanos , Lactente , Masculino , Variações Dependentes do Observador , Reprodutibilidade dos Testes , Estudos Retrospectivos , Adulto Jovem
11.
Eur J Med Genet ; 62(6): 103531, 2019 Jun.
Artigo em Inglês | MEDLINE | ID: mdl-30142436

RESUMO

Interstitial deletions involving chromosome region 6p21.31p21.2 have not been previously reported in the literature. Here, we present a 2 year old girl with global developmental delay, severe speech delay, dysmorphic features, laryngeal cleft, anterior descending aorta that occluded the left main bronchus and a novel de novo deletion of chromosome 6: arr[hg19] 6p21.31p21.2 (35462950-36725083)x1. The deletion, which was diagnosed by array comparative genomic hybridization and further confirmed with fluorescence in situ hybridization, was approximately 1.26 Mb and contained 28 RefSeq genes. The deleted region includes 24 protein coding genes and 4 non-coding genes. This represents a novel microdeletion that has not been previously reported in the literature.


Assuntos
Aorta Torácica/anormalidades , Deleção Cromossômica , Transtornos Cromossômicos/genética , Cromossomos Humanos Par 6/genética , Anormalidades Congênitas/genética , Deficiências do Desenvolvimento/genética , Laringe/anormalidades , Pré-Escolar , Transtornos Cromossômicos/patologia , Anormalidades Congênitas/patologia , Deficiências do Desenvolvimento/patologia , Feminino , Humanos , Laringe/patologia , Síndrome
12.
Pediatr Emerg Care ; 35(3): e47-e48, 2019 Mar.
Artigo em Inglês | MEDLINE | ID: mdl-29232350

RESUMO

Diagnosis and treatment of laryngeal clefts (LCs) particularly type I pose a challenge. Although rare, type I LCs are becoming increasingly identified in recent years, and this is perhaps due to both an increased awareness and better diagnostic modalities. We report a young infant presenting with feeding difficulty and respiratory distress related to LC. The pertinent literature is also reviewed.


Assuntos
Anormalidades Congênitas/diagnóstico , Laringoscopia/métodos , Laringe/anormalidades , Obstrução das Vias Respiratórias/etiologia , Transtornos da Alimentação e da Ingestão de Alimentos/etiologia , Humanos , Lactente , Masculino , Respiração Artificial/métodos
13.
Int J Pediatr Otorhinolaryngol ; 118: 59-61, 2019 Mar.
Artigo em Inglês | MEDLINE | ID: mdl-30583194

RESUMO

OBJECTIVE: To report on the prevalence of voice disturbances in pediatric airway patients. METHODS: Consecutive patients seen in a specialized Center for Airway Disorders at a tertiary children's hospital from February 2017 to September 2017 were included. Patients' families were invited to complete a pediatric voice health handicap index (pVHI) questionnaire. Patients underwent evaluation including flexible laryngoscopy and/or direct laryngoscopy and bronchoscopy. RESULTS: 146 patients were included. Of these children, 73 patients (50.3%) presented with swallowing difficulty and 44 patients (30.3%) presented with respiratory complaints. Only 9 patients (6.2%) reported hoarseness initially. The median age at referral was 2.7 years of age (interquartile range: 1.4-4.3). The mean total pVHI score was 9.5 (± 12.9). Sixty-seven patients (45.9%) had abnormal pVHI findings of score > 4. Six patients (4.1%) had pVHI > 40. The mean pVHI score was 26.0 (± 21.1) among 12 patients with a history of tracheostomy, 12.0 (± 14.3) among 30 patients with laryngeal cleft, and 9.0 (± 9.9) among 19 patients with laryngomalacia. CONCLUSION: Voice disturbances are not uncommon in pediatric patients evaluated for airway disorders. Although patients may present with primary concerns for breathing or swallowing difficulties, many of these patients may need further work-up and treatment for dysphonia.


Assuntos
Transtornos de Deglutição/epidemiologia , Disfonia/epidemiologia , Rouquidão/epidemiologia , Broncoscopia , Pré-Escolar , Anormalidades Congênitas/epidemiologia , Feminino , Humanos , Lactente , Laringomalácia/epidemiologia , Laringoscopia , Laringe/anormalidades , Masculino , Prevalência , Sistema Respiratório , Índice de Gravidade de Doença , Inquéritos e Questionários , Traqueostomia/estatística & dados numéricos
14.
Int J Pediatr Otorhinolaryngol ; 116: 159-163, 2019 Jan.
Artigo em Inglês | MEDLINE | ID: mdl-30554689

RESUMO

INTRODUCTION: Interarytenoid injection augmentation at the time of initial diagnostic endoscopy for aspiration and dysphagia may result in near-immediate improvement in swallowing function, potentially obviating the need for future formal endoscopic repair of type 1 laryngeal cleft. Interarytenoid injection augmentation may also address physiologic aspiration. Early treatment of type 1 laryngeal cleft may allow for expedited liberalization of feedings. The objective of this study was to evaluate the effect of interarytenoid injection augmentation (IIA) for type 1 laryngeal clefts (LC-1) on short-term swallowing function assessed by videofluoroscopic swallowing study (VFSS). METHODS: This was a retrospective cohort study of patients age ≤24 months with dysphagia on preoperative VFSS who underwent IIA with calcium hydroxyapatite for LC-1 during direct laryngoscopy and bronchoscopy from June to October 2017 at a tertiary care academic subspecialty hospital. Exclusion criteria included prior endoscopic or open LC repair (n = 1), gastrostomy tube dependence (n = 1), additional procedures at the time of IIA (supraglottoplasty, frenulectomy, n = 1). Children without postoperative VFSS within 30 days of injection were excluded (n = 2). Fifteen children met inclusion criteria for analysis. The primary endpoint was improvement in safely swallowed consistency as defined by recommendation to liberalize diet by at least a half-consistency (e.g. half-honey to nectar thick liquid). Secondary endpoints included clinical assessment of dysphagia and postoperative respiratory events. RESULTS: Median [range] age at injection was 15.2 [7.7-24.3] months and 67% of patients were female (n = 10). The majority (13/15) of patients were full-term and 80% of patients (n = 12) had documented gastroesophageal reflux disease (GERD). Median time from injection to VFSS was 16 [9-29] days. Improvement in safely swallowed consistency was noted in 60% (n = 9) of patients. Aspiration completely resolved in two patients. Swallow function was unchanged in 40% of patients (n = 6); no patients experienced worsening dysphagia. No respiratory complications were documented during inpatient observation. CONCLUSION: IIA is a safe procedure that may result in immediate improvement in dysphagia in select patients with LC-1. IIA does not address neurologic, developmental, or other anatomic etiologies of dysphagia. Additional studies are required to determine long-term efficacy of IIA on dysphagia and pulmonary complications, as well as the patient- and caregiver-related outcome measures.


Assuntos
Materiais Biocompatíveis/administração & dosagem , Anormalidades Congênitas/terapia , Transtornos de Deglutição/terapia , Deglutição/fisiologia , Durapatita/administração & dosagem , Laringe/anormalidades , Cartilagem Aritenoide , Broncoscopia/métodos , Pré-Escolar , Estudos de Coortes , Transtornos de Deglutição/etiologia , Feminino , Fluoroscopia/métodos , Humanos , Lactente , Laringoscopia/métodos , Laringe/efeitos dos fármacos , Laringe/cirurgia , Masculino , Estudos Retrospectivos , Resultado do Tratamento
15.
Medicine (Baltimore) ; 97(51): e13709, 2018 Dec.
Artigo em Inglês | MEDLINE | ID: mdl-30572501

RESUMO

RATIONALE: Congenital high airway obstruction syndrome (CHAOS) is defined as complete or partial obstruction of the fetal upper airways. Laryngeal atresia is the most frequent cause. PATIENT CONCERN: A male neonate born with poor reactions, weakly spontaneous breathing and cyanosis of the limbs was referred to our hospital. DIAGNOSIS: CHAOS with tracheoesophageal fistula. INTERVENTION: A tracheostomy was performed and a 3.0-mm internal diameter tracheostomy tube was inserted. OUTCOMES: Neonatal survival depended on our immediate postnatal intervention. LESSONS: In summary, a multidisciplinary team including otolaryngologists, radiologists, obstetricians, and anesthesiologists must be present during the whole diagnosis and treatment process.


Assuntos
Obstrução das Vias Respiratórias/congênito , Obstrução das Vias Respiratórias/diagnóstico , Fístula Traqueoesofágica/complicações , Fístula Traqueoesofágica/diagnóstico , Obstrução das Vias Respiratórias/complicações , Obstrução das Vias Respiratórias/cirurgia , Diagnóstico Diferencial , Evolução Fatal , Humanos , Recém-Nascido , Laringe/anormalidades , Masculino , Fístula Traqueoesofágica/cirurgia , Traqueostomia
17.
Artigo em Chinês | MEDLINE | ID: mdl-30293271

RESUMO

Laryngeal clefts as exceptionally rare congenital anomalies have been reported more and more recently. The clinical symptoms and presentations base on degree of clefts and variety of comorbidities. Mild types of clefts may have minimal clinical impact, whereas severe types may be life-threatening and even lead to death before diagnosis. With more attention of these anomalies and advances in endoscopy, the diagnosis, evaluation and surgery of laryngeal clefts have acquired great improvement, especially International Pediatric Otolaryngology Group(IPOG)have reported consensus guidelines on the diagnosis and management of type Ⅰ laryngeal clefts in 2017. This review of laryngeal clefts may be beneficial to decrease missed diagnosis or misdiagnosis and to achieve individual-based treatment.


Assuntos
Anormalidades Congênitas/diagnóstico , Anormalidades Congênitas/cirurgia , Laringe/anormalidades , Criança , Consenso , Erros de Diagnóstico/prevenção & controle , Humanos , Laringoscopia , Laringe/cirurgia , Otolaringologia
18.
Int J Pediatr Otorhinolaryngol ; 115: 10-18, 2018 Dec.
Artigo em Inglês | MEDLINE | ID: mdl-30368367

RESUMO

OBJECTIVES: To improve the recognition of differences in presentation amongst patients with type 1 laryngeal clefts of various ages and better understand the age dependent outcomes of injection laryngoplasty. A second aim was to analyze the discrepancies between swallow assessment modalities in various age groups with type I laryngeal clefts undergoing injection laryngoplasty. METHODS: A retrospective review of electronic medical records of patients who underwent injection laryngoplasty from 2009 through 2015 at a tertiary care children's hospital. Data extracted included: Demographics, histories and physical exam findings, diagnostic studies, and medical and surgical treatments. RESULTS: Most (72/102, 70.6%) patients were male with a median gestational age at birth of 37 weeks (range 24-41 weeks). Formula thickening and GERD medications were used in 94/102 (92.2%) and 97/102 (95.1%) patients, respectively. Comorbid GERD, laryngomalacia, tracheomalacia, and subglottic stenosis were present in 98/102 (96.1%), 40/102 (39.2%), 9/102 (8.8%), and 14/102 (13.7%) patients, respectively. There was no significant difference in demographics, comorbidities or medical therapy between age groups. Symptoms at presentation differed between age groups with stridor (χ2(1) = 11.6, p = 0.002) and cyanosis (χ2(1) = 8.13, p = 0.012) being more common in the 0-3-month group compared to the 12-36 month group. Symptom resolution and the odds of undergoing additional surgery (second injection or suture repair) over time, however, did not differ. There was a significant reduction in aspiration with thins during FEES (McNemar χ2(1) = 10.7, p = 0.002) and aspiration with nectar during MBS (McNemar χ2(1) = 5.26, p = 0.035) post-injection. After injection, there was significant agreement in aspiration with thins between FEES and MBS (kappa = 0.308 ±â€¯SE 0.170, p = 0.035). However, finding aspiration with thins was more common during MBS than during FEES (McNemar χ2(1) = 7.00, p = 0.016). There were no differences in swallow evaluation findings between the age groups. CONCLUSIONS: Symptoms of type I laryngeal clefts may differ by age. However, there was no impact of age on the safety and efficacy of surgical intervention.


Assuntos
Anormalidades Congênitas/cirurgia , Transtornos de Deglutição/cirurgia , Deglutição , Laringoplastia/métodos , Laringe/anormalidades , Fatores Etários , Sulfato de Bário , Pré-Escolar , Anormalidades Congênitas/fisiopatologia , Meios de Contraste , Transtornos de Deglutição/diagnóstico por imagem , Transtornos de Deglutição/etiologia , Endoscopia Gastrointestinal , Feminino , Humanos , Lactente , Recém-Nascido , Injeções , Laringe/fisiopatologia , Laringe/cirurgia , Masculino , Reoperação , Aspiração Respiratória/etiologia , Estudos Retrospectivos , Resultado do Tratamento
19.
JAAPA ; 31(11): 36-40, 2018 Nov.
Artigo em Inglês | MEDLINE | ID: mdl-30358678

RESUMO

Stridor is a high-pitched respiratory sound that signals upper airway obstruction. It can be encountered by clinicians in a variety of clinical settings and requires a team-based, interdisciplinary approach. Early recognition is crucial, as the differential diagnosis can be broad, and causes range from benign to life-threatening. This article reviews the most commonly encountered causes of chronic congenital stridor in infants, focusing on the diagnostic approach, pathophysiology, clinical presentation, and management strategies.


Assuntos
Anormalidades Congênitas/etiologia , Laringomalácia/complicações , Laringe/anormalidades , Sons Respiratórios/etiologia , Paralisia das Pregas Vocais/complicações , Doença Crônica , Anormalidades Congênitas/diagnóstico , Anormalidades Congênitas/terapia , Diagnóstico Diferencial , Diagnóstico Precoce , Refluxo Gastroesofágico/etiologia , Humanos , Recém-Nascido , Laringomalácia/epidemiologia , Laringoestenose/complicações , Equipe de Assistência ao Paciente , Sons Respiratórios/diagnóstico , Traqueomalácia/complicações , Paralisia das Pregas Vocais/epidemiologia
20.
Int J Pediatr Otorhinolaryngol ; 114: 134-137, 2018 Nov.
Artigo em Inglês | MEDLINE | ID: mdl-30262351

RESUMO

We report a bizarre case of accessory larynx in an infant with OEIS syndrome (omphalocele, cloacal exstrophy, imperforated anus & spinal defects). This is the first reported case in literature of a duplicate accessory larynx which is a mirror image of the true larynx. A congenital duplication of the larynx is a rare anomaly and can present in various forms. In this case, the infant presented with recurrent lung infection and inability to wean off oxygen. Scope revealed severe laryngomalacia in addition to the accessory larynx. Hence, supraglottoplasty was done with aim to resolve the lung and airway problem.


Assuntos
Anus Imperfurado/complicações , Hérnia Umbilical/complicações , Doenças da Laringe/diagnóstico , Laringe/anormalidades , Escoliose/complicações , Anormalidades Urogenitais/complicações , Anormalidades Múltiplas , Feminino , Humanos , Recém-Nascido , Doenças da Laringe/complicações , Doenças da Laringe/cirurgia , Laringoscopia/métodos , Laringe/cirurgia , Sons Respiratórios/etiologia
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