Management of congenital subglottic hemangioma.

OBJECTIVE: This study was conducted to assess the safety and efficacy of individualized management of congenital subglottic hemangioma (CSH) at the Children's Memorial Hospital in Chicago; describe treatment modalities, including endoscopic excision with the carbon dioxide (CO2) laser, systemic corticosteroids, and intralesional corticosteroid injection with short-term intubation; and determine the success of these various strategies in avoiding tracheotomy. METHODS: During the 10-year period between January 1, 1988 and December 31, 1997, 28 infants were diagnosed with CSH. A retrospective review of medical records was undertaken to determine demographics, presenting symptoms, location of the lesion, therapeutic modality, and complications. All patients were contacted at the time of writing. RESULTS: Twenty-eight patients met the criteria for this study. Gender distribution was 1.8:1 female to male. Age at diagnosis ranged from 4 weeks to 8 months, with a mean of 78.8 days and a median of 60 days. The location of CSH was most often posterior and on the left. Associated hemangiomas were found in 14 (50%) cases. The most common symptoms were stridor and cough. Management included 1 to 13 operative direct laryngoscopies and bronchoscopies, endoscopic excision with the (CO2) laser, and the use of systemic and intralesional corticosteroids. No patient required tracheotomy. CONCLUSION: Morbidity and the need for tracheotomy in CSH patients can be minimized using a combination of therapeutic modalities. Each infant's treatment is individualized based on the severity of the patient's symptoms and the morphology of the lesion.     

Does early expansion surgery have a role in the management of congenital subglottic stenosis?

To better define the clinical course of congenital subglottic stenosis, the authors reviewed a cohort of 27 children managed with long-term tracheotomy and interval bronchoscopy. Rates of spontaneous resolution were 19% at 6 months, 39% at 12 months, and 69% at 18 months (Kaplan-Meier survival analysis). Children older than 4 months of age at tracheotomy tended to have more rapid resolution of their stenoses (P = .08). Overall, the median time to decannulation was 15.8 months (95% confidence interval, 13.9 to 17.6 months). One death was possibly tracheotomy-related, and 12 (44%) of the children experienced complications or sequelae resulting in new or extended hospitalization. Considering the prolonged cannulation necessary for spontaneous resolution of congenital subglottic stenosis, the role of long-term tracheotomy as the sole form of management may be limited. Once the need for intervention is apparent, the authors recommend early consideration of cricoid split or laryngotracheoplasty as alternatives to long-term tracheotomy.     

Minimally invasive endoscopic management of subglottic stenosis in children: Success and failure

Objective

To assess the efficacy and safety of endoscopic management of subglottic stenosis both as a primary and as an adjunctive treatment in the pediatric population.

Methods

Retrospective review of pediatric patients with subglottic stenosis undergoing endoscopic airway procedures at a tertiary care pediatric medical center. Outcomes were assessed by systematic review to determine the success and failure of the endoscopic approach.

Results

Forty patients (22 male, 18 female) underwent endoscopic interventions for a diagnosis of subglottic airway stenosis between 2003 and 2006. Age ranged from 22 days old to 20 years old. Recorded degree of subglottic stenosis ranged from 10% to 99%. Fifty-three percent (21/40) had a history of prematurity, and 40% (16/40) had secondary airway diagnoses. Twenty-four patients underwent an endoscopic intervention initially (including laser or dilation, with or without topical mitomycin treatment), including four patients who underwent tracheostomy prior to the first endoscopic intervention. Sixteen underwent laryngotracheoplasty initially, including ten patients who underwent tracheostomy prior to the laryngotracheoplasty. Endoscopic treatment resulted in resolution of symptoms, and/or decannulation, and no further need for an open procedure in 58% of patients. Of the 24 patients undergoing endoscopic interventions initially, 14 patients underwent two or more endoscopic interventions, and 10 patients subsequently required tracheostomy or laryngotracheoplasty. When endoscopic procedures were used as an adjunct to laryngotracheoplasty, 60% (12/20) had resolution of symptoms, underwent decannulation, and did not require tracheostomy or revision laryngotracheoplasty.

Conclusions

The endoscopic approach can be successful in the management of properly selected patients with subglottic stenosis, either as the initial treatment modality or as an adjunctive treatment in cases of re-stenosis after open airway surgery. The likelihood of success with a minimally invasive procedure as the primary treatment decreases with worsening initial grade of subglottic stenosis.     

婴儿声门下血管瘤治疗初探

目的初步探讨婴儿声门下血管瘤的不同治疗方法及疗效。方法回顾性分析我院自2008～2011年收治的声门下血管瘤患儿13例,7例气管切开术后分次行平阳霉素瘤体内注射,4例口服普萘洛尔治疗,1例口服泼尼松片治疗,1例单纯行气管切开姑息治疗。结果 7例平阳霉素注射治疗,血管瘤完全消退,6例成功拔管,平均疗程7.5个月;4例口服普萘洛尔,3例血管瘤完全消退,1例大部分消退,平均疗程4.8个月;1例口服泼尼松片后8个月声门下肿块大部分消退,伴发库欣综合征;1例单纯气管切开患儿,于3年后血管瘤完全消退,拔除气管套管。结论平阳霉素瘤体内注射疗效是肯定的,但绝大多数患儿需行气管切开,且每次注药均需住院全麻下进行,疗程长。普萘洛尔可短时间内使瘤体缩小、改善呼吸道梗阻症状,避免了手术创伤性操作,且副作用小,可作为一线药物使用。     

婴幼儿先天性声门下血管瘤的诊断和治疗

目的：探讨婴幼儿先天性声门下血管瘤的诊断和治疗。方法：9例患儿,反复喉喘鸣、喂哺困难及喉梗阻发作,电子支气管镜检查确诊为先天性声门下血管瘤。7例为声门下单侧病变,2例为双侧病变。其中8例采用气管切开后血管瘤内平阳霉素注射法进行治疗,术后患儿带金属气管套管出院,2周后门诊随访电子支气管镜复查,如果肿瘤未消失,再次行平阳霉素注射,直至肿瘤完全消失后进行堵管及拔管;1例单侧声门下血管瘤行瘤体吸割术,未行气管切开。结果：平阳霉素治疗的6例声门下单侧病变患者经过1次治疗后血管瘤完全消失,2例双侧病变患者经过2次治疗后血管瘤完全消失,8例均顺利堵管及拔管。1例行声门下血管瘤吸割术患者,术后2周复查黏膜光滑,完全修复。结论：对于体积较大,阻塞声门下腔范围较广的血管瘤,采用气管切开后血管瘤内平阳霉素注射法,能迅速解除气道梗阻,缩短自然病程,且戴管时间短,无术后声门下狭窄,是一种安全有效的治疗措施。对于体积较小的声门下血管瘤,在麻醉配合良好的情况下,行吸割术并给予良好止血,可避免气管切开,起到微创效果。     

Subglottic hemangiomas in infants: treatment with intralesional corticosteroid injection and intubation

Six children with a subglottic hemangioma were treated in the Sophia Children's Hospital in the period 1982-1987 by means of intralesional corticosteroid injection, followed by intubation. After treatment all children were symptomfree. In 3 patients this result was attained after 1 injection, in 2 after 3 injections. One patient needed 5 injections. The average duration of intubation was 19 days (7-36). Three months (1/2-7 1/2) after the onset of therapy all patients were free of symptoms. No serious complications were observed. No patient needed a tracheotomy. The average follow-up period was 3.3 years (1 1/2-5 1/2). The authors feel that not only the effect of intralesional corticosteroids, but also local gentle pressure by the tube is of therapeutic importance. The above-mentioned treatment of subglottic hemangioma in children is now the treatment of choice in our clinic.     

Use of potassium titanyl phosphate (KTP) laser in management of subglottic hemangiomas

OBJECTIVE: to study the role of KTP laser in management of subglottic hemangioma. DESIGN: retrospective analysis of patients with subglottic hemangioma treated by the senior authors. Setting: tertiary care teaching hospital. PATIENTS: twelve patients with subglottic hemangiomas. INTERVENTION: patients were treated with KTP laser (eight cases), CO(2) laser (two cases) or observation (two cases). MAIN OUTCOME MEASURE: resolution of symptoms, decrease in size of subglottic hemangioma or tracheotomy decannulation. RESULTS: All patients treated with KTP laser or CO(2) laser had resolution of symptoms and five tracheotomy-dependent children were decannulated. CONCLUSION: subglottic hemangioma is a potentially life-threatening disease seen in young children. Most authors recommend use of either CO(2) or open surgical excision. There is very little data available on the use of KTP lasers in the management of subglottic hemangiomas. The KTP laser beam is preferentially absorbed by hemoglobin making this laser system more applicable to the treatment of vascular tumors such as the hemangioma. KTP laser is a good tool for management of subglottic hemangioma with a low incidence of complications.     

Subglottic hemangioma treated with interferon alpha 2A

Infantile subglottic hemagioma is a rare vascular malformation involving the subglottic larynx and although present from birth, symptoms will not be noted until later in infancy (due to tendency to enlarge). Typically presents with a progressive crouplike illness that begins a few weeks after birth and the infant develops inspiratory stridor, which becomes expiratory as the obstruction increases. Although benign lesions which involute spontaneously, they may demand the attention of an ENT surgeon to maintain a secure airway. We report a case of a 3-month-old female patient (full term, normal delivery) who was referred to us for investigation of respiratory distress. Endoscopic examination revealed a hemangioma at the left lateral wall of the subglottic larynx while the rest of the airway was normal. The infant was treated with a combination of steroids (dexamethazone 0.5 mg x 3 per os) and interferon A-2a (650.000 IU subcutaneously, every other day, for 12 months) and had fast improvement of her symptoms. Repeated endoscopy 3 months after the diagnosis reveal impressive remission of the subglottic hemangioma. Throughout the years, a variety of treatments have been proposed and utilised for subglottic hemangioma. Interfron 2-alpha, drug acting by interference with angiogenesis, is very effective in treating subglottic hemangiomas without the need for tracheostomy. Its side effects are generally not serious. As congenital subglottic hemangiomas have tendency towards spontaneous regression, conservative treatment seems more appropriate compared to more aggressive treatment that carry substantial risks of long term complications.     

Propranolol for the treatment of subglottic hemangiomas

IntroductionInfantile subglottic hemangiomas are rare causes of airway obstruction. They begin to proliferate at 1–2 months of age and can cause biphasic stridor with or without respiratory distress. Diagnosis requires direct visualization by direct laryngoscopy and bronchoscopy. Various therapeutic options have been utilized for treatment, including tracheotomy, open surgical excision, laser ablation, intralesional steroid injection, systemic steroids, and now oral propranolol.MethodsWe present a retrospective chart review of infantile subglottic hemangiomas over a 5-year span (January 2005–2010) at a tertiary care pediatric hospital. IRB approval was obtained, and charts were reviewed to find patients with subglottic hemangiomas, including patient characteristics, presentation, workup, medical and surgical management, and outcomes. A case presentation demonstrates diagnostic, management, and treatment strategies and dilemmas encountered.ResultsNine patients were found to have infantile subglottic hemangiomas. Six of nine patients were treated with laser excision, with five of the six having localized subglottic hemangiomas. In 2009, three of four patients were initiated on propranolol as first-line treatment; the fourth had comorbidities which precluded this. Of the three, two showed improvement, while a third, who also had bearded hemangioma, required tracheotomy.DiscussionInfantile subglottic hemangiomas are rare but essential in the differential diagnosis of biphasic stridor. Although propranolol has been effective in treating cutaneous and airway hemangiomas, our experience suggests that this is not consistent for subglottic hemangiomas. In an area where airway compromise can be lethal, we must extend caution and monitor these patients closely as they may require adjuvant therapy.     

The application of the potassium-titanyl-phosphate (KTP) laser in the management of subglottic hemangioma

BACKGROUND: Subglottic hemangioma is the most common neoplasm of the infant airway. Most lesions involute spontaneously; however, some may grow to cause life-threatening respiratory tract distress. The standards of treatment have been tracheotomy, corticosteroids, and laser vaporization. However, use of the carbon dioxide laser has been associated with increased risk of damage to adjacent mucosa and an increased risk for the development of subglottic stenosis postoperatively. OBJECTIVE: To review our experience with the use of the potassium-titanyl-phosphate laser in the treatment of subglottic hemangioma. PATIENTS AND METHODS: A retrospective review of 6 patients with subglottic hemangioma treated with the potassium-titanyl-phosphate laser was carried out at a tertiary care children's hospital. Patients' medical charts were evaluated for factors such as age, sex, degree of airway obstruction, location of hemangioma, number of laser procedures performed, and postoperative results, including short- and long-term complications. RESULTS: All 6 patients had significant relief of airway obstruction after use of the potassium-titanyl-phosphate laser. Five of the patients had localized disease, and one had circumferential subglottic involvement. The average number of procedures was 1.7. There were no intraoperative complications. One patient had prolonged intubation following surgery, and only one developed grade 1 subglottic stenosis requiring dilation. Long-term results have been promising in that all patients are asymptomatic and follow-up laryngoscopy and bronchoscopy have shown complete resolution of the hemangioma. CONCLUSION: The potassium-titanyl-phosphate laser can be used to provide significant clinical relief of airway obstruction due to grade 1 and 2 subglottic hemangioma, with minimal complications.     

Paediatric subglottic stenosis

Most reports on paediatric subglottic stenosis have arisen from tertiary referral centres. At the Sheffield Children's Hospital, referrals come from the surrounding region such that almost all paediatric airway problems from this area are seen. This allows an opportunity to look at the overall picture of this condition. A retrospective study was performed to identify all children with subglottic stenosis requiring a tracheostomy since 1979 and to examine the management and eventual outcome in relation to the severity of their stenosis. Forty-two children were identified. There have been four mortalities, two of which have been tube related. Twenty-two laryngotracheoplasty procedures have been performed in 17 patients. Successful decannulation has been achieved in 82%. In the majority of children with more minor stenoses conservative management has lead to successful decannulation. However, with increasingly severe stenosis the necessity of undergoing a laryngotracheoplasty procedure to provide an adequate airway becomes more likely.     

Management of subglottic stenosis in infancy and childhood

During the 12-year period between June 1987 and June 1999, 141 children underwent curative treatment for subglottic laryngeal stenosis at La Timone Children’s Hospital in Marseille, France. Ninety-six children (68%) were under the age of 5 years; 106 (75%) presented with acquired stenosis and 93 had narrowing involving over 70% of the subglottic lumen. Endoscopic laser surgery was performed in 25 cases and open surgery in 116. Open surgical techniques included laryngotracheoplasty with autologous cartilage interposition in 83 cases, laryngotracheal split in 22, and cricotracheal resection in 11. After decannulation, 132 children (94%) were able to breathe normally through the upper airway. Perspectives for development of new techniques and improvement of conventional methods are discussed. Received: 10 November 1999 / Accepted: 16 December 1999     

Controlled risk of stenosis after surgical excision of laryngeal hemangioma

OBJECTIVE: To evaluate the risk of subglottic stenosis after surgical excision of congenital subglottic hemangioma. DESIGN: Retrospective analysis and case series. SETTING: Tertiary care teaching hospital. PATIENTS: A total of 13 pediatric patients diagnosed with subglottic hemangioma with unilateral, bilateral, or circular lesions and more than 50% airway obstruction between 1992 and 2001. INTERVENTION: Open surgical excision was performed as a single-stage procedure either as primary or secondary intention. The cricoid cartilage was left open at the end of the procedure. Postoperative intubation was carried out in a pediatric intensive care unit. MAIN OUTCOME MEASURE: An adequate airway after surgical excision. RESULTS: All patients were successfully extubated. No recurrence was noted. Three patients developed subglottic stenosis, two grade 1 and one grade 2. All 3 showed a favorable outcome and did not require reintubation. One needed endoscopic management of the stenosis. Of these 3 cases, 2 occurred after carbon dioxide laser treatment (out of 3) and 1 after circumferential dissection (out of 3). CONCLUSIONS: Extubation after surgery was successful in all cases of subglottic hemangioma. Risk of subglottic stenosis was limited and occurred only after circumferential dissection, especially if associated with prior traumatic laser damage of the hemangioma.     

Primary cricotracheal resection in children: indications, technique and outcome

OBJECTIVE: Treatment of subglottic stenosis has benefited greatly from development of grafting techniques such as larygontracheoplasty. Meanwhile, cricotracheal resection of the stenotic area and a major part of the cricoid cartilage have been shown to give excellent results in adults. PATIENTS AND METHODS: From June 1993 to June 2000, we performed cricotracheal resection, as the primary surgical procedure, for acquired (n=13), congenital (n=2) or mixed (n=l) subglottic stenosis in 16 pediatric patients comprising seven boys and nine girls. At the time of surgery, the mean weight was 16 kg and the mean age was 5 years. Degree of stenosis was classified as grade II in one case, grade III in 12, and grade IV in three. Eight patients underwent two-stage procedures with postoperative tracheostomy. A rolled reinforced silastic stent was placed for a mean period of 20 days and the tracheostomy tube was removed within a mean period of 44 days after stent removal. Eight patients underwent single-stage procedures. The Portex endotracheal tube was used for a mean period of 4 days. RESULTS: All patients underwent regular clinical and endoscopic postoperative examination. Mean follow-up was 38 months. No interference with laryngotracheal growth has been noted in any case, including the five with follow-ups longer than 5 years. CONCLUSIONS: The indications for laryngotracheoplasty and cricotracheal resection in children with subglottic stenosis are still unclear. Decisions must be made on a case-by-case basis. In this article the authors discuss decisional factors in terms of clinical findings, surgical techniques, potential complications, and outcome.     

Microdebrider removal under suspension laryngoscopy: An alternative surgical technique for subglottic hemangioma

Objective

To describe microdebrider removal of subglottic hemangiomas and evaluate the efficacy of this procedure.

Methods

This retrospective study was conducted at an otorhinolaryngology department. Seven consecutive patients with subglottic hemangiomas who failed to respond to pharmacologic treatment were treated by microdebrider removal under suspension laryngoscopy. Respiratory symptoms and hemangioma status were assessed preoperatively and postoperatively.

Results

The mean age of patients at disease onset was 68 days (range, 24–120 days), and their mean age at the time of surgical treatment was 132 days (range, 55–195 days). Their initial pharmacologic treatment included steroids, propranolol, and/or antibiotics. The mean percentage of airway obstruction was initially 36% (range, 30–50%), and this value changed to 61% (50–80) before surgery. Microdebrider removal of hemangiomas was successful in all patients without any intraoperative or postoperative complication. The mean operative time was 30 min (range, 21–47 min). Three patients required orotracheal intubation for 12 or 24 h after surgery, and no reintubation or tracheotomy was required in this series. Symptoms such as stridor and inspiratory retraction were resolved approximately 72 h after surgery, but wheezing was generally resolved at a later time point. One year later, no respiratory symptom was noted in these patients, and endoscopic examination revealed a sufficient glottis opening.

Conclusions

Surgical removal with a microdebrider is advantageous as a routine surgical technique for small and moderate pediatric subglottic hemangioma.     

Recurrent croup presentation, diagnosis, and management

PURPOSE: The lack of clinical insight into recurrent croup often leads to underdiagnosis of an upper airway lesion, and subsequently, inadequate treatment. This study examined the underlying etiology, diagnosis, treatment, and clinical outcome of patients with a history of recurrent croup identified at initial presentation. The aim was to present common diagnostic features and suggest new diagnostic and management recommendations. MATERIALS AND METHODS: A retrospective chart review of 17 children diagnosed with recurrent croup. Demographic, historical, and intraoperative data as noted in clinic charts were collected. Specific collected data included age, sex, chief complaint, presenting symptoms, past medical history, previous medication history, number of emergency room visits and inpatient admissions, tests/procedures performed and corresponding findings, current treatment given, and posttreatment clinical outcome. RESULTS: Six (35.3%) patients presented initially with a past medical history of gastroesophageal reflux disease. Fourteen (82.3%) patients had positive endoscopic evidence of gastroesophageal reflux. For these 14 patients, 44 laryngopharyngeal reflux lesions were noted, with 32 (72.7%) occurring in the subglottis. All 14 patients demonstrated various degrees of subglottic stenosis ranging from 30% to 70% (Cotton-Myer grade I-II). All 17 patients (100%) demonstrated subglottic stenosis ranging from 15% to 70% airway narrowing. CONCLUSIONS: History suggestive of recurrent croup requires close monitoring and expedient direct laryngoscopy/bronchoscopy for diagnosis. Long-term follow-up and antireflux treatment are necessary as well as endoscopic documentation of significant reflux resolution.     

Surgical treatment of infantile subglottic hemangioma

Three cases of infantile subglottic hemangioma are described. Generally conservative management is favored by most authors. These present lesions were removed surgically by midline cricotracheotomy. Follow-up showed that all children were well and without complaints. The authors believe that besides management with the CO2 laser and short-term steroid therapy, operative treatment of infantile subglottic hemangioma is a valuable alternative which should receive more attention. All children were decannulated a few days after surgery.     

The anterior cricoid split: the Children's Hospital of Philadelphia experience

The anterior cricoid split procedure (ACS) is an alternative to tracheotomy in selected infants with subglottic stenosis. From February 1983 to October 1987, 26 children underwent ACS at the Children's Hospital of Philadelphia. All children had subglottic stenosis. Six children had previously undergone tracheotomy. A successful outcome, allowing extubation or decannulation, correlated only with decreased pre-operative intubation time and was achieved in 73% of these patients. A failed ACS had little adverse affect on the subsequent management of these children.     

Subglottic stenosis in children: a conservative approach

Subglottic stenosis may be either congenital or acquired and is one of the commonest causes of acute and chronic airways obstruction in neonates and infants. Most acquired stenoses result from perinatal intubation. The management of these lesions remains controversial, particularly regarding the timing of surgical intervention. At Alder Hey Children's Hospital a series of 33 patients with subglottic stenosis have been reviewed. A conservative approach, employing long-term tracheotomy as the mainstay of treatment, has achieved a success rate of 72.7%, and only 3 patients have so far needed corrective surgery (9%), of which 1 has been decannulated to date. One patient (3%) died of bronchopulmonary dysplasia. We believe that, where close follow-up is possible, a conservative approach is the method of choice.     

Pediatric tracheotomy: 17 year review

OBJECTIVE: To study the outcomes, complications, and indications for pediatric tracheotomies performed at a major tertiary care children's hospital, Starship Children's Hospital in Auckland, New Zealand, over the period 1987-2003. METHODS: A retrospective review of hospital records from 1987 to 2003 was conducted to assess all pediatric patients who had undergone tracheotomies. RESULTS: A total of 122 tracheotomies (119 surgical, 3 percutaneous) were performed on patients less than 16 years of age. Upper airway obstruction (including craniofacial dysmorphism, n=40, and subglottic stenosis, n=18) was the most common indication for surgery (n=86; 70%) with a lesser number (n=36; 30%) requiring tracheotomy for prolonged ventilation. The median age at tracheotomy was 4.5 months in patients with upper airway obstruction and 16 months in those requiring prolonged ventilation. Decannulation was carried out successfully in 92 patients (75%), although 6 (6.5%) subsequently required recannulation. The overall complication rate was 51% (n=62). Early postoperative complications occurred in a total of 9 (7.4%) patients, including difficulties with ventilation in intensive care due to inadequate seal or tube position in 5 (4.1%), and accidental decannulation in 3 (2.5%). Late complications included localized granulation in most patients, for which 15 (12.3%) required intervention whilst under a routine planned general anesthetic. Major vascular erosion was not encountered in any patient, although 5 (4.1%) required intervention for minor bleeding associated with granulation tissue. Suprastomal collapse occurred in 13 patients (10.7%); but did not affect their subsequent decannulation, although 2 (1.6%) developed tracheotomy-related subglottic stenosis. Closure of tracheocutaneous fistulas was required in 16 (13.1%) decannulated patients. Only 2 patients (1.6%) died from tracheotomy-related complications, with an overall mortality rate of 14%. CONCLUSIONS: Pediatric tracheotomies performed at Starship Children's Hospital between 1987 and 2003 were associated with a low incidence of procedure-related mortality and morbidity and successful decannulation in most cases. The majority of procedures were performed to treat upper airway obstruction, most commonly caused by craniofacial dysmorphism or subglottic stenosis.