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Purpose To describe a series of children with extensive PNF or treatment refractory PLGG treated on a compassionate basis with trametinib. Methods We report on six patients with NF‐1 treated with trametinib on a compassionate basis at British Columbia Children's Hospital since 2017. Data were collected retrospectively from the patient record. RAPNO and volumetric criteria were used to evaluate the response of intracranial and extracranial lesions, respectively. Results Subjects were 21 months to 14 years old at the time of initiation of trametinib therapy and 3/6 subjects are male. Duration of therapy was 4–28 months at the time of this report. All patients had partial response or were stable on analysis. Two patients with life‐threatening PNF had a partial radiographic response in tandem with significant clinical improvement and developmental catch up. One subject discontinued therapy after 6 months due to paronychia and inadequate response. The most common adverse effect (AE) was grade 1–2 paronychia or dermatitis in 5/6 patients. There were no grade 3 or 4 AEs. At the time of this report, five patients remain on therapy. Conclusion Trametinib is an effective therapy for advanced PNF and refractory PLGG in patients with NF‐1 and is well tolerated in children. Further data and clinical trials are required to assess tolerance, efficacy and durability of response, and length of treatment required in such patients., Here, we report on response to Trametinib, a MEK inhibitor in children with NF‐1. Our experience presented in this report demonstrates that Trametinib is an effective therapy for advanced, life‐threatening PNF, and treatment‐refractory PLGG in patients with NF‐1 and is well tolerated in children.

Create a competency-based assessment tool for pediatric esophagoscopy with foreign body removal.Blinded modified Delphi consensus process.Tertiary care center.A list of 25 potential items was sent via the Research Electronic Data Capture database to 66 expert surgeons who perform pediatric esophagoscopy. In the first round, items were rated as "keep" or "remove" and comments were incorporated. In the second round, experts rated the importance of each item on a seven-point Likert scale. Consensus was determined with a goal of 7 to 25 final items.The response rate was 38/64 (59.4%) in the first round and returned questionnaires were 100% complete. Experts wanted to "keep" all items and 172 comments were incorporated. Twenty-four task-specific and 7 previously-validated global rating items were distributed in the second round, and the response rate was 53/64 (82.8%) with questionnaires returned 97.5% complete. Of the task-specific items, 9 reached consensus, 7 were near consensus, and 8 did not achieve consensus. For global rating items that were previously validated, 6 reached consensus and 1 was near consensus.It is possible to reach consensus about the important steps involved in rigid esophagoscopy with foreign body removal using a modified Delphi consensus technique. These items can now be considered when evaluating trainees during this procedure. This tool may allow trainees to focus on important steps of the procedure and help training programs standardize how trainees are evaluated.5. Laryngoscope, 131:1168-1174, 2021.

Objectives/hypothesis Create a competency-based assessment tool for pediatric esophagoscopy with foreign body removal. Study design Blinded modified Delphi consensus process. Setting Tertiary care center. Methods A list of 25 potential items was sent via the Research Electronic Data Capture database to 66 expert surgeons who perform pediatric esophagoscopy. In the first round, items were rated as "keep" or "remove" and comments were incorporated. In the second round, experts rated the importance of each item on a seven-point Likert scale. Consensus was determined with a goal of 7 to 25 final items. Results The response rate was 38/64 (59.4%) in the first round and returned questionnaires were 100% complete. Experts wanted to "keep" all items and 172 comments were incorporated. Twenty-four task-specific and 7 previously-validated global rating items were distributed in the second round, and the response rate was 53/64 (82.8%) with questionnaires returned 97.5% complete. Of the task-specific items, 9 reached consensus, 7 were near consensus, and 8 did not achieve consensus. For global rating items that were previously validated, 6 reached consensus and 1 was near consensus. Conclusions It is possible to reach consensus about the important steps involved in rigid esophagoscopy with foreign body removal using a modified Delphi consensus technique. These items can now be considered when evaluating trainees during this procedure. This tool may allow trainees to focus on important steps of the procedure and help training programs standardize how trainees are evaluated. Level of evidence 5. Laryngoscope, 131:1168-1174, 2021.

We report on a family with three siblings, male and female, affected by congenital bilateral limitation of vocal cord abduction, with the additional finding of clubfeet in two. The paternal family history suggests an autosomal dominant inheritance. The siblings and father also have mild craniofacial features, which may be an expression of variability or may be unrelated. The association between congenital vocal cord paralysis and clubfeet has been reported with additional major features or in the context of Charcot-Marie-Tooth disease. However, the two in isolation have only been reported in one other family previously. Genomic analyses of the family, including chromosomal microarray and exome sequencing, showed neither a likely pathogenic variant in a known disease gene nor a compelling candidate gene variant. We propose that the association of these two findings constitutes a novel recognizable phenotype, for which a genetic cause remains undetermined.

Objective: To present a novel case of Luc’s Abscess with coexisting intracranial abscess and venous sinus thrombosis. Case Presentation: A 4-year-old male had fever for two weeks, left-sided otalgia and otorrhea for 1 week and then suddenly developed left temporal soft tissue swelling and papilledema. Computerized tomography scan revealed a 3.5 x 2.3 x 1.2cm subperiosteal temporal space (Luc’s) Abscess, a 4.6 x 2.7 x 2.5 cm temporal lobe abscess, and transverse plus sigmoid sinus thrombosis. The patient was successfully treated with surgical drainage, long-term intravenous antibiotics, and anti-coagulation. Conclusion: To our knowledge, this is the first documented case of Luc’s abscess with a coexisting intracranial abscess and/or venous sinus thrombosis. Although Luc’s abscess is a rare and usually relatively benign complication of acute otitis media, early diagnosis and treatment remain essential in preventing potentially life-threatening disease progression.

Tracheal-bronchus is an aberrant bronchus arising from the lateral tracheal wall, superior to the carina. A "False-carina" can be classified as a sub-type. This clinical entity will be defined and the clinical presentation, diagnosis and management of five patients with variations of the anatomical definition of a False-carina, identified at our institution, will be reviewed. Congenital bronchial abnormalities, including False-carina, have important implications in the overall management of the airway. Management can range from expectant in asymptomatic patients to surgical intervention in cases of recurrent respiratory infections. Awareness and understanding of this clinical entity allows for timely investigation, diagnosis and appropriate intervention.

Objectives To develop (1) a practical and comprehensive algorithm of instruments and techniques available to treat neonates with tracheobronchial foreign bodies, based on the patient's subglottic diameter and (2) primary and secondary prevention strategies for neonatal tracheobronchial foreign bodies. Methods (1) Analysis of the case of a severely premature infant who presented with the incidental radiological finding of a 2 cm suction catheter tip, which, over a two week period, had migrated between her main bronchi; the foreign body was removed with a previously unreported combination of instruments: a 3 French (F) flexible urological forceps through the side port of a 2.5 mm rigid bronchoscope. (2) In vitro testing of typical foreign bodies and readily available endoscopic instruments. Results We have developed a practical and comprehensive algorithm for the treatment of neonates with tracheobronchial foreign bodies, as well as primary and secondary prevention strategies. Conclusions The treatment algorithm and prevention strategies may reduce morbidity and mortality from neonatal tracheobronchial foreign bodies.

Summary Cystic fibrosis (CF) affects between 1 in 2000 and 1 in 4500 children of European descent [R.S. Mendelsohn, B.M. Cohen. Otorhinolaryngologic aspects of cystic fibrosis. Archives of Otolaryngology 79 March (1964) 312–17]. There have been few reported cases of fungal sinus infections in patients with CF [S.K. Wise, T.T. Kingdom, L. McKean, J.M. DelGaudio, G. Venkatraman. Presence of fungus in sinus cultures of cystic fibrosis patients. American Journal of Rhinology 19 January–February (1) (2005) 47–51]. We report a unique case of a young male with CF presenting with severe destructive sinonasal polyposis, which was refractory to all treatment until he was treated for a concomitant fungal bronchopulmonary infection.

Summary Objective : To report a case of a 17-month-old female presenting with a giant retropharyngeal abscess and to outline the minimally invasive technique that was used to drain it. Design : Case study. Results : A previously healthy 17-month-old female was found by contrast-enhanced computed tomography (CECT) to have a retropharyngeal abscess (RPA) which extended from the level of the nasopharynx to the level of the aortic arch. The RPA measured 4.5cm transversely, 2.7cm antero-posteriorly and 8.0cm cephalocaudally. After orotracheal intubation, transoral incision and drainage of the abscess was performed. Based on the CECT findings, the baby Yankauer suction was advanced 6cm beyond the oropharyngeal incision (within the retropharynx) to break the distal septae and completely drain the RPA with a minimally invasive technique. Discussion: Our case describes the management of the largest RPA in the medical literature. We present the first report of the use of a baby Yankauer suction advanced through the oropharyngeal incision to drain intrathoracic RPA loculations.

Summary We present two cases of foreign body aspiration that occurred during the 2004–2005 Christmas season. In one 8-month-old boy, the hardware from a Christmas tree ornament became a supraglottic foreign body. In another 10-month-old boy, a metallic sticker in the shape of a Christmas tree traveled from the glottis to the nasopharynx and back to the glottis. The winter holidays are a time when children may have less supervision and greater exposure to potential aerodigestive foreign bodies. It is especially important that parents are vigilant and that physicians are perceptive of the symptoms of foreign body aspiration, especially during the holiday season.

Objectives To review clinical presentations and management strategies for children with plastic bronchitis. Methods Retrospective chart review. Results Seven patients required rigid bronchoscopy to remove bronchial casts over a 17-year study period. Mean age at presentation was 60 months. Mean follow-up was 53 months. Co-morbidities included: congenital heart disease (n = 3), chronic pulmonary disorders (n = 2) and sickle cell disease (n = 1). 4 patients required multiple bronchoscopies for recurrent casts. Adjunctive topical therapies were administered in all 7 patients, without complication. Rigid bronchoscopy for cast removal was performed in 2 patients who were on extra-corporal membrane oxygenation (ECMO), using special precautions to safeguard the ECMO catheters. Conclusions Bronchial casts in children may present acutely or sub-acutely. Recurrent casts are unusual; however, in combination with severe cardiac disease may lead to mortality. Adjunctive topical therapies are still under investigation. Special safeguards for ECMO catheters are imperative. This case series complements and adds to the International Plastic Bronchitis Registry.

Summary Objective: To report two cases in which toddlers drank a caustic liquid which their parents had been using to prepare free-base and crack cocaine. Design: Case series. Results: Two toddlers presented with first- and second-degree burns of the oral cavity, pharynx and esophagus from caustic ingestion. The first had drunk ammonia from an unmarked clear plastic container the morning after “a party at a friend's house”. The second had drunk potassium hydroxide from a clear plastic container which was labelled “pH UP” and had a small “Danger” sign. Both children were treated with intravenous hydration, steroids, antibiotics and analgesics. Fortunately, neither child suffered long-term physical sequelae. When questioned, the parents of both children admitted to using these alkalis to prepare free-base and crack cocaine. Appropriate steps were taken by social services to protect these children from further neglect. Discussion: Despite federal legislation and public education, pediatric caustic ingestion remains a significant problem. These cases suggest that, in certain cases of pediatric caustic ingestion, caregivers should be questioned regarding possible drug abuse. Larger warning labels and further public education might help prevent many cases of pediatric caustic ingestion.

Background : The lingual tonsil is an often overlooked source of morbidity in older children. Case histories : (1) An adolescent male with Trisomy 21 presented with an intense fear of swallowing solid food after a severe choking event. Flexible endoscopy revealed asymmetric hypertrophy of his lingual tonsil. A videofluoroscopic swallow study revealed altered hypopharyngeal swallowing mechanics, with temporary trapping of food in the lingual tonsil. The patient also had obstructive sleep apnea (OSA). CO 2 laser lingual tonsillectomy was performed in two stages with complete symptom resolution and no morbidity. (2) An adolescent female presented with recurrent mild hemoptysis, which eluded diagnosis for 2 years. Eventually, a bleeding site was discovered within an inflamed lingual tonsil. The patient had also developed loud snoring, odyntophagia, and globus sensation. CO 2 laser lingual tonsillectomy was performed in one stage with complete symptom resolution and no morbidity. Conclusions : These are the first cases reported in which the lingual tonsils were found to be the cause of choking in the pediatric population or pseudo-hemoptysis at any age. CO 2 laser lingual tonsillectomy is a safe and effective procedure, which has been facilitated by recent advances in CO 2 laser technology.

Purpose : To illustrate the challenge and approach to diagnosing a laryngeal foreign body (FB) in a child with pre-existing laryngeal pathology. Methods : Case report. The diagnosis of laryngeal FB was initially missed in a 21-month old-infant. Two weeks later, the child experienced worsening in symptoms. Results : Flexible laryngoscopy revealed the presence of a plastic object in the subglottis, which was then removed under general anesthesia. Conclusions : For children with pre-existing laryngeal pathology, a sudden unexplained worsening of laryngeal symptoms, particularly dysphonia and biphasic stridor, should prompt the clinician to consider the possibility of a thin, sharp laryngeal FB.

Importance Because tonsillar enlargement can have substantial ill health effects in children, reliable monitoring and documentation of tonsil size is necessary in clinical settings. Tonsil grading scales potentially allow clinicians to precisely record and communicate changes in tonsil size, but their reliability in a clinical setting has not been studied. Objective To assess the interobserver and intraobserver reliability of the Brodsky and Friedman tonsil size grading scales and a novel 3-grade scale. Design, Setting, and Participants Cross-sectional study between June 2012 and August 2013 at a tertiary pediatric otolaryngology outpatient clinic at British Columbia Children's Hospital. We recruited 116 children, aged 3 to 14 years, with no major craniofacial abnormalities. For each child, 2 separate tonsil assessments (with at least a 5-minute interval in between) were conducted by 4 independent observers: 2 staff pediatric otolaryngologists, 1 otolaryngology trainee (fellow or resident), and 1 medical student. Each observer assessed and graded tonsil sizes using 3 different scales. Main Outcomes and Measures Interobserver and intraobserver reliabilities were assessed by deriving the intraclass correlation coefficients (ICCs) and Pearson correlation coefficients, respectively. To discount for any asymmetric scores, all data analysis was conducted on the left tonsil measurement only. Results Mean interobserver reliability was highest for the Brodsky grading scale (ICC, 0.721; Cronbach α, 0.911), followed by the Friedman grading scale (ICC, 0.647; Cronbach α, 0.879) and the 3-grade scale (ICC, 0.599; Cronbach α, 0.857). The mean intraobserver reliabilities for the Brodsky, Friedman, and modified 3-grade scales were 0.954, 0.932, and 0.927, respectively. Conclusions and Relevance The Brodsky grading scale offered the highest interobserver and intraobserver reliability when compared with the Friedman and novel 3-grade scales. The results of this study would support the uniform use of the Brodsky scale for future clinical and research work.

Summary We report our experience, not previously reported in the literature, with a young male Jehovah Witness who aspirated a metal button backing into his right bronchus intermedius. While in the operating room, a size 5, 30 cm Storz rigid ventilating bronchoscope with a 0 degrees rod lens telescope was passed through the larynx to visualize the bronchus intermedius. Then, using the “duckbill” telescopic alligator forceps, the sharp edges of the foreign body were grasped and removed. The patient recovered well and was discharged home hours after without any complications.

Congenital midline cervical cleft (CMCC) is a rare developmental defect of the anterior neck normally characterized by an atrophic mucosal plaque with a cranial nipple-like skin tag, a short caudal sinus, and may be attached to a subcutaneous fibrous cord of variable length. Clinically, patients present at an early age with, white females being the most commonly affected population. In addition to aesthetic concerns, CMCC can prevent full extension of the neck, result in micrognathia and torticollis, predispose patients to infection, and can coexist with other clefting defects or cysts. Fewer than 50 cases have been published in the English-language literature. Herein, we report a case of CMCC that also presented with a mild contracture of the right sternohyoid muscle. The embryopathogenesis, histopathology, diagnosis, and treatment of this rare condition are also discussed.

To evaluate the effectiveness of the BC Children's Hospital (BCCH) Safe Start Choking Prevention campaign, which began in 2002 and used newspaper, magazine, radio, and television interviews of parents, injury prevention experts, and physicians to educate the public about choking risks, prevention, and treatment (CRPT); to compare our campaign with other campaigns; and to determine if a new strategy is needed to educate the public about CRPT.A retrospective analysis of the number of cases of laryngeal, bronchial, and esophageal foreign bodies (LBEFBs) that required rigid endoscopy under general anesthesia at BCCH and/or admission to BCCH was conducted. Data from January 1997 to December 2002 to January 2003 to December 2006 were compared. The world literature on choking prevention and analysis of barriers to CRPT public education was reviewed.The data demonstrate the ineffectiveness of our traditional media campaign. The number of total operating room visits at BCCH for LBEFBs increased marginally after our choking prevention campaign began. Published reports from Israel and Crete indicate that educational campaigns that included direct teaching of CRPT by otolaryngologists and other health educators to parents and their children are effective; however, such campaigns would be difficult to replicate within larger populations.A new strategy for CPRT public education is required. We are creating an animated video and a comprehensive, interactive website to teach CRPT to preteens, teenagers, and adults (with prospective validation of knowledge transfer and long-term outcome measurement).

Objective:We present an extremely rare case of severe intracranial hypertension secondary to sigmoid sinus compression by a group A streptococcal epidural abscess.Method:Case report and review of the world literature.Results:A five-year-old boy was treated for acute otitis media and group A streptococcal bacteraemia, but subsequently developed severe intracranial hypertension. Computed tomography revealed that, although the sigmoid sinuses were not thrombosed, the patient had a dominant right sigmoid sinus that was almost completely compressed by a small epidural abscess. After surgical decompression of the epidural abscess, with aggressive debridement of the granulation tissue from the sigmoid sinus wall, the patient awoke from general anaesthesia with complete resolution of his symptoms and signs of intracranial hypertension. He suffered no sequelae over the subsequent six months' follow up.Conclusion:This is the first reported case of intracranial hypertension due to an epidural abscess causing sigmoid sinus compression without thrombosis. This case illustrates the fact that, even in the absence of thrombosis of the sigmoid sinus, a small epidural abscess may require urgent surgical treatment.

To describe the importance of imaging with the use of magnetic resonance (MR) or computed tomography (CT) during the diagnostic workup of a patient with sensorineural hearing loss to determine the status of the cochlear nerve.Retrospective review.Tertiary care academic institution.A pediatric database was used to find patients with diagnoses of absent cochlear nerve. A retrospective chart review was performed from July 1999 to July 2004 to evaluate the route to diagnosis and any concomitant factors. Patients who had presented at kindergarten screening were included. Review was made of the audiologic investigations undertaken: routine audiometry, auditory brainstem response (ABR), and distortion product otoacoustic emission (OAE). Radiologic investigations were also reviewed consisting of CT and/or MR scans.The database yielded 12 cases of cochlear agenesis. Four patients were excluded because they had absence of the entire inner ear structures ipsilateral to the aplastic cochlear nerve (two cases) and because they had multiple congenital anomalies (two cases). There were equal numbers of males and females. There was a slight left-sided preponderance (5:3), and ages ranged from 5 through 7 years. All children had failed the initial screening audiogram. Follow-up audiologic evaluation revealed either profound loss or dead ear or a failed ABR in the presence of normal OAE testing. All patients had internal auditory canals less than 1.4 mm or MR-compatible findings.Agenesis of the cochlear nerve may be more common than previously thought, especially in an otherwise healthy, nonsyndromic, school-aged child. Although audiometric evaluation alone usually strongly suggests the diagnosis, definitive evaluation with MR remains the gold standard.

To report our experience at British Columbia's Children's Hospital (BCCH) with the use of methylene blue (MB) to demarcate preauricular sinuses (PASs) and branchial sinuses and fistulae (BSF) in children under general anesthesia just prior to surgical excision.A retrospective chart review was performed of all cases at BCCH between March 2003 and April 2006 in which MB was used to demarcate PASs and BSF. A review of the literature regarding the utility and safety of topical MB was performed.MB demarcation of 20 PASs enabled precise resection with the involved auricular cartilage and obviated the need for en bloc resection. In 11 BSF, MB differentiated between sinuses and fistulae, identified the branchial cleft involved, demonstrated (in two patients) a thin fistula that was not visible on high-resolution contrast-enhanced computed tomography, and enabled excision with the use of small incisions and minimal dissection. In our series, there have been no recurrences and no complications from the use of MB.MB demarcation of PASs and BSF enables minimally invasive surgery and helps ensure complete resection. This is an extremely useful and relatively safe technique that has been underreported in the otolaryngology literature.

To determine when it is safe for healthy children to return to home, school, and sports after uncomplicated electrosurgical adenoidectomy (EA).(1) A survey of Canadian otolaryngologists (COs) regarding their EA complication rates and their opinions regarding best practice standards for patient care after EA. (2) Informal retrospective analysis of approximately 200 children who have been allowed to resume sports 3 days after uncomplicated EA.(1) Thirteen COs responded to the survey. The total estimated EA cases performed were 8617 over 10 years. Four patients were known to have bled mildly; none required reoperation. Eleven of 13 COs supported discharge home 2 hours after uncomplicated EA if other standard discharge criteria were met. Seven of 13 COs supported a return to school the next day if the child had returned to his or her usual state of health; however, 2 COs suggested 7 to 10 days at home. Four of 13 COs supported a return to sports 3 days after uncomplicated EA. (2) None of the 200 children who had been allowed to resume sports 3 days after uncomplicated EA have had any known bleeding. At least 10 toddlers have actually engaged in unplanned strenuous exercise within 4 to 6 hours of EA without adverse effect.Considerable variation exists in Canada in terms of current practice standards regarding return to home, school, and sports after uncomplicated EA. Survey data suggest that, after uncomplicated EA, it is safe for healthy children to return home after 2 hours and to school the next day. Preliminary data suggest that it may be safe for children to resume sports 3 days after uncomplicated EA; however, ideally, this should be studied prospectively, on a large scale.

Cystic lymphatic vascular malformations are benign lesions that can cause disfigurement and functional impairment. Complete surgical resection is often difficult, and clinical recurrence is common. Sclerotherapy has been used as an alternative to excision. OK-432 is a lyophilized mixture of Streptococcus pyogenes and benzylpenicillin which, when injected into a lesion, has shown significant ability to reduce its size or obliterate it completely.The authors report a series of 12 patients treated in this fashion at the Vascular Anomalies Clinic, British Columbia Children's Hospital, between 1999 and 2004. All patients underwent imaging of the lesion: 10 had magnetic resonance imaging, one had a computed tomographic scan, and one had ultrasound examination. Six patients had macrocystic malformations (cystsor = 2 cm) and six had microcystic or combined lymphaticovenous malformations. Patients were treated with intralesional injection of OK-432. The position of the injection was confirmed by angiography and/or ultrasound in 10 cases. Response to treatment was assessed clinically.All patients with macrocystic malformations had complete resolution or good response to treatment. None required any additional treatment. In contrast, those with microcystic or combined malformations responded poorly. All of these patients underwent subsequent excision without adverse consequences. The size and location of the lesion did not correlate with response to treatment. Seventy-five percent of patients experienced pyrexia. Local swelling is an expected phenomenon and must be anticipated, particularly for lesions near the airway.OK-432 is an excellent treatment for patients with macrocystic lymphatic malformations. However, it is ineffective for microcystic lesions.

Summary Objective To review the demographics, pathophysiology and management of aspiration of sharp, metallic foreign bodies; to review prevention strategies. Design Case series and review of the related literature (1932–2006). Setting Tertiary care pediatric hospital. Patients All patients presenting to BC Children's Hospital with aspiration of sharp, metallic foreign bodies since 1998. Results The seven patients ranged in age from 11 to 15 years (mean 13 years). The two boys had been playing with other boys at the time of aspiration. One aspirated a thumbtack and one aspirated a homemade blowdart (which traveled from his carina to his subglottis with coughing). Three of the five girls had been holding thumbtacks or a darting pin between their lips while putting up posters or sewing. The other two girls, who aspirated thumbtacks, refused to provide a history. One was treated successfully 6 days after the aspiration. The other girl withheld the history for over a year, presented with hemoptysis and eventually required thoractomy and right main bronchotomy to remove the tack. All of the other patients were diagnosed and successfully treated by rigid bronchoscopy within 24 h. (Two patients initially had a failed attempt at foreign body removal by flexible bronchoscopy at another hospital.) This is the second largest series of aspiration of sharp, metallic foreign bodies since at least 1966. Adolescents are still aspirating sharp, metallic foreign bodies because of the same behaviors described by Jackson and Jackson in 1932. Education about choking hazards is needed for this age group. Conclusions Aspiration of a sharp, metallic foreign body is a serious injury which is best treated by rigid bronchoscopy. These injuries potentially could be prevented through education.

A case of aspiration of an extracted molar is presented. The main objective of this paper is to provide dental colleagues with an educational framework on foreign-body aspiration to help prevent delayed diagnosis of such events in the future.

OBJECTIVE A comparison between the literature and our management of pediatric patients presenting with otogenic lateral sinus thrombosis. DESIGN A retrospective case series of five pediatric patients. SETTING Four patients were treated at BC Children's Hospital, whereas the fifth patient was treated in New Westminster, BC. All were treated between 1994 and 2001. METHODS A retrospective chart review was conducted with a literature review for otogenic lateral sinus thrombosis. MAIN OUTCOME MEASURES Treatment success was based on resolution of acute infection and neurologic symptoms. RESULTS Five patients, four males and one female, aged 2 to 14 years were reviewed. Three patients were treated successfully without mastoidectomy. One patient received a mastoidectomy that yielded no pus or granulation tissue within the mastoid cavity. One patient required a mastoidectomy after failure to respond to bilateral myringotomy and tympanostomy tube insertion. Although no pus was seen in the mastoid cavity, perisinus pus was found after unroofing the sigmoid sinus plate; free flow of blood was obtained on needle aspiration of the sinus, and the sinus was not surgically opened. CONCLUSION The current literature states that the management of otogenic lateral sinus thrombosis includes high-dose intravenous antibiotics with a mastoidectomy and possible opening of the sinus. In our retrospective case series, three of five patients recovered completely without mastoidectomy, and a fourth had a mastoidectomy deemed to have been unnecessary. We conclude that intravenous antibiotics and insertion of a tympanostomy tube are sufficient treatment for selected cases of otogenic lateral sinus thrombosis. Mastoidectomy with possible opening of the sinus should be reserved for patients refractory to the above conservative treatment.

Background Noninflammatory masses of the salivary gland region in children are extremely rare. Therefore, very few published individual and institution-based experiences exist. Design Retrospective chart review from 1990 through 1997. Setting University-based children's hospital. Design Patients 18 years of age or younger with a tumor in the salivary gland region. Masses of infectious origin were excluded. Hemangiomas and lymphangiomas were tallied for relative incidences only. Results Three hundred twenty-four consecutive cases of salivary gland masses were found: 192 hemangiomas (59.2%), 89 lymphangiomas (27.5%), and 43 (13.3%) solid masses. No significant difference was found between the age at presentation of the patients with benign solid tumors and the patients with malignant solid tumors (mean + SEM age, 7.2 + 0.7 years). Sixty-one percent of the masses were found in the parotid region; 18% were localized to the submandibular gland region; and the remaining 21% were located in a minor salivary gland site. The most common benign perisalivary masses were pilomatrixomas (20.9%), followed by pleomorphic adenomas (11.6%). The most common malignant masses were mucoepidermoid carcinomas (9.3%), followed by rhabdomyosarcomas (7.0%). Treatment was individualized to the disease. Twenty-two patients had adequate data for follow-up analysis (mean + SEM follow-up, 30.0 + 8.4 months). Four patients (18.2%) experienced recurrent or residual disease and were alive with disease at last follow-up, and 100% of our population demonstrated disease-specific survival at last follow-up. Conclusions Vascular lesions outnumber solid tumors of the salivary gland region. The most common salivary tumors were pleomorphic adenomas, followed by mucoepidermoid carcinomas. Although certain solid salivary masses may demonstrate locally aggressive behavior, the overall prognosis is favorable.

A retrospective chart review was performed to quantify the postoperative complications and outcomes of 82 consecutive cases of laryngotracheal reconstruction (LTR) and cricotracheal resection (CTR) performed at a pediatric tertiary care hospital over the last 9 years. Six cases of respiratory syncytial virus (RSV) bronchiolitis and 8 cases of cervical pseudomonal wound abscess (PWA) were identified in a total of 12 patients. All of these infections occurred after single-stage LTR or CTR. Both RSV bronchiolitis and PWA were associated with significantly more unexpected days of intubation and admission to the intensive care unit, as well as higher rates of failure of LTR. Ossification of the cricoid cartilage, grade IV subglottic stenosis, and untreated gastroesophageal reflux disease (GERD) were also associated with restenosis. Trisomy 21 did not significantly influence the success rate of pediatric LTR. Both RSV bronchiolitis and PWA are potentially preventable complications of pediatric LTR and CTR. We propose strategies to prevent these infections. We also advocate the treatment of GERD during the healing phase of LTR.

Background Cystic hygromas are characterized by a proliferation of small vessels and lymphatics with intervening fibrous tissue. Studies have shown malignant tumors and some benign neoplasms are dependent on angiogenesis, the induction of new capillaries from preexisting vessels. Growth and progression of these tumors are associated with a disturbance in the balance of angiogenic inducers and inhibitors. We have postulated that cells derived from cystic hygromas are angiogenic due to secretion of higher levels of angiogenic inducers that promote vascular proliferation. Design A large cystic mass was surgically removed and a portion of the sterile tumor was immediately placed in the medium. The tissue was minced, washed in phosphate-buffered saline, and grown to near confluence. Conditioned medium was collected under serum-free conditions after 48 hours. Secreted proteins were concentrated, quantitated, and analyzed in an in vitro endothelial cell migration assay and by Western blot. Antibody to factor VIII-related antigen was performed to confirm endothelial cell origin of the cultured cells. Main Outcome Measures In vitro angiogenic activity of secreted proteins in a capillary endothelial migration assay was tested by using blocking antibodies to angiogenic inducer, basic fibroblast growth factor, and angiogenic inhibitor, thrombospondin-1. Total protein levels of thrombospondin-1 were determined by Western blot. Results Cells isolated from cystic hygroma are angiogenic in vitro and this angiogenic activity is due to secretion of high levels of angiogenic inducer, basic fibroblast growth factor, and lower levels of naturally occurring angiogenic inhibitor, thrombospondin-1. Conclusions Cystic hygromas may represent another neoplasm dependent on angiogenesis. The angiogenic activity is due in part to elevated levels of potent angiogenic inducer, basic fibroblast growth factor. Anti-angiogenic therapy directed at the endothelial cell may help suppress the growth of cystic hygromas.

Importance To our knowledge, the present study is the first double-blind, randomized, placebo-controlled trial in children to compare nasal preparation sprays administered before flexible nasendoscopy with placebo. Objective To compare the degree of pain experienced by children undergoing flexible nasendoscopy after 1 of 3 intranasal sprays: placebo, decongestant with topical local anesthetic (TLA), or decongestant without TLA. Design, Setting, and Participants A randomized placebo-controlled trial with blinding of participants, caregivers, observers, and otolaryngologists was conducted in a tertiary pediatric otolaryngology ambulatory clinic. Participants included a consecutive sample of children aged 3 to 12 years requiring flexible nasendoscopy. Exclusion criteria included concomitant respiratory tract infection, known allergy to a trial agent, or previous flexible nasendoscopy. One hundred fifty-one children were assessed for eligibility; 24 eligible children refused participation and 69 were included and block-randomized. All completed the study, and there were no adverse events. Interventions Nasal spray administration of placebo (normal saline); xylometazoline hydrochloride, 0.05% (decongestant); or lidocaine hydrochloride, 1%, with xylometazoline hydrochloride, 0.05% (TLA with decongestant) was performed 10 minutes before flexible nasendoscopy. Main Outcomes and Measures Primary outcome measure was the child-reported Wong-Baker Faces Pain (WBFP) scale. Secondary outcomes included the caregiver-proxy WBFP scale; the Face, Legs, Activity, Cry, and Consolability (FLACC) scale; and the physician-reported Difficulty of Procedure Visual Analog Scale (DPVAS). Results Twenty-three children were recruited in each of the intervention arms. Baseline characteristics were comparable between groups. The mean child-rated WBFP scale scores were 2.4, 1.8, and 2.2 for the placebo, decongestant, and TLA with decongestant groups, respectively ( P = .45). Although the finding was statistically nonsignificant, decongestant had the lowest mean caregiver-proxy WBFP scale score, lowest observer-rated FLACC scale score, and highest physician-rated DPVAS score. Subgroup analysis did not demonstrate any correlation between the outcomes and age or sex. Conclusions and Relevance This study revealed no statistically significant difference in the discomfort experienced by children undergoing flexible nasendoscopy after placebo, decongestant, or TLA with decongestant. Decongestant was associated with the least discomfort (on child, caregiver, and observer-rated pain scale scores) and the lowest rating for difficulty of procedure. With these findings, the study suggests that there is no significant benefit of topical decongestant with or without TLA compared with placebo in reducing pain associated with pediatric flexible nasendoscopy. Trial Registration clinicaltrials.gov Identifier:NCT01351298

Objectif: Le but de cette etude etait de determiner la precision des tests au diapason de Rinne et Weber dans la prediction du degre de perte d'audition dans l'otite moyenne avec epanchement (OME) chez l'enfant en le comparant avec l'audiometrie tonale. Devis: Une etude prospective a l'aveugle a ete faite chez des enfants entre 2 et 11 ans avec des OME unilaterales ou bilaterales. Localisation: Les patients ont ete recrutes a partir de la clinique d'Oto-rhino-laryngologie pediatrique du British Columbia's Children's Hospital, un centre tertiaire a Vancouver au Canada. Methode: Nous avions comme sujets, 58 enfants avec OME unilaterale et bilaterale. Les tests de Rinne et Weber ont ete faits en utilisant le diapason a 256 Hz et 512 Hz. Une audiometrie tonale a permis d'etablir la presence et le degre de Rinne audiometrique (ecart aerien-osseux EAO). Variables evaluees: Les resultats du diapason ont ete mis en correlation avec L'EAO pour en determiner l'exactitude. Resultats: Le test de Weber ne montre pas d'association entre L'EAO et la reponse de lateralisation en utilisant soit le diapason de 256 Hz (p=.484) ou celui de 512 Hz (p = .120). Pour le test de Rinne, une augmentation de L'EAO etait significativement associee a un Rinne inverse utilisant autant le diapason de 256 Hz que celui de 512 Hz fourchette (p

Objectif: Determiner quand il est sans danger pour les enfants de retourner a la maison, a l'ecole et de faire du sport suite a une adenoidectomie a l'electrocautere non compliquee (AE). Devis: (1) Un sondage aupres des oto-rhino-laryngologistes canadiens (OC) concernant leurs taux de complications relies a l'AE et leurs opinions sur les meilleurs standards de pratique pour les soins des patients apres une AE. (2) Une analyse informelle retrospective sur 200 enfants qui ont pu retourner a leurs activites sportives 3 jours apres une AE. Resultats: (1) Treize OC ont repondu au sondage. Nous estimons a 8,617 sur 10 ans le nombre de cas evalues. Quatre patients ont saigne moderement; aucun n'a eu besoin d'etre reopere. Onze des 13 OC sont d'accord pour le retour a la maison 2 heures apres une AE sans complication et en repondant a d'autres criteres. Sept des 13 OC sont d'accord pour un retour a l'ecole le jour apres l'intervention si l'enfant a retrouve son etat de sante; cependant, deux OC ont suggere de 7 a 10 jours a la maison. Quatre des 13 OC sont d'accord pour un retour aux sports 3 jours apres l'AE sans complication. (2) Aucun des 200 enfants qui sont retournes a leurs activites sportives 3 jours apres une AE n'a eu de saignements portes a notre attention. Au moins 10 enfants entre 18 et 36 mois sont retournes sans problemes a des activites vigoureuses non planifiees quatre a 6 heures apres leur AE. Conclusion: Des variations considerables existent au Canada en terme de pratiques actuelles concernant le retour a la maison, a l'ecole et aux sports apres une AE sans complication. Les donnees du sondage suggerent qu'apres une AE sans complication, il est securitaire pour des enfants en sante de retourner a la maison apres 2 heures et a l'ecole le lendemain. Les donnees preliminaires suggerent qu'il est securitaire pour les enfants de retourner a leurs activites sportives 3 jours apres l'intervention; cependant il serait ideal d'etudier ces recommandations prospectivement a plus grande echelle.

Objectif: Les objectifs de cette etude etaient de (1) examiner les tendances recentes dans la demographie et la presentation clinique des enfants consultant au British Columbia's Children's Hospital pour aspiration d'un corps etranger et (2) de developper des recommandations quant aux noix et autres produits du meme type dans l'alimentation des enfants. Methode: Revue de dossier et revue de la mecanique de la deglutition chez les enfants. Resultats: Nous avons procede a 51 bronchoscopies rigides pour suspicion de corps etranger chez des enfants de moins de 3 mois entre juillet 1997 et juillet 2001. De ce nombre, 27 (53%) avaient moins de 18 mois. Dans ce dernier groupe, un temoin a vu l'episode d'etouffement dans 24 (89%) des cas et chez 22 (81%) on a retrouve un corps etranger. Les noix, carottes crues et grains de mais souffle representent 14 (64%) des corps etrangers aspires chez ces enfants. Avant 2 ans, les enfants sont mal equipes pour croquer et avaler de la nourriture dure et croustillante puisqu'ils n'ont pas de deuxiemes molaires et qu'ils s'ajustent encore a la descente de leur larynx. Conclusion: Le taux d'aspiration de corps etranger est alarmant en Colombie Britannique. La majorite de ces cas auraient pu etre prevenus par une meilleure information de la population. Les parents et educateurs devraient etre informes que les enfants de moins de 3 ans ne devraient jamais manger de noix et autres aliments durs et croustillants. Nous recommandons une campagne d'information publique.

To assess the effect of endothelium-derived relaxing factor (EDRF) on diaphragmatic vascular resistance at rest and during contractions, we studied an in situ isolated diaphragm preparation in anesthetized and mechanically ventilated dogs. The arterial supply of the left diaphragm (phrenic artery) was catheterized and perfused with arterial blood at a fixed flow rate. Drugs were infused through a side port of the arterial catheter at 1/100th of the phrenic arterial flow. The inferior phrenic vein was catheterized to complete the isolation from the systemic circulation. Three sets of experiments were performed. In set 1 (n = 3), we infused endothelium-dependent (acetylcholine, ACh) and endothelium-independent (sodium nitroprusside, SNP) dilators at increasing concentrations. ACh and SNP infusion elicited a dose-dependent decline in phrenic vascular resistance (Rphr) at concentrations greater than 10(-8) M and 0.50 micrograms/ml, respectively. In set 2 (n = 15), we infused an inhibitor of EDRF synthesis and release, L-argininosuccinic acid (ArgSA), at increasing concentrations (10(-4), 3 x 10(-4), and 6 x 10(-4) M). ArgSA produced a dose-dependent increase in Rphr. Infusion of another EDRF inhibitor (NG-nitro-L-arginine, LNA, 6 x 10(-4) M) elicited increase in Rphr similar to that induced by ArgSA. In set 3 (n = 25), we infused ArgSA or LNA (6 x 10(-4) M) simultaneously with ACh and SNP and during sustained (2-Hz) contractions of the diaphragm. Both ArgSA and LNA completely reversed ACh vasodilation, whereas SNP vasodilation was reversed by 26 and 11%, respectively. ArgSA or LNA infusion during contractions reversed vasodilation by 48 and 52%, respectively.(ABSTRACT TRUNCATED AT 250 WORDS)