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 biology and management of subglottic hemangioma: past, present, future

OBJECTIVES/HYPOTHESIS: Objectives were 1) to review the presentation, natural history, and management of subglottic hemangioma; 2) to assess the affect of five variables (age, gender, degree of subglottic narrowing, location and extent of subglottic hemangioma, and lack or presence of other hemangioma) and the outcome of six different treatment modalities (conservative monitoring, corticosteroid, laser surgery, tracheotomy, laryngotracheoplasty, and interferon) in the management of subglottic hemangioma; and 3) to present specific guidelines to help determine the best possible treatment modality at the time of initial presentation. STUDY DESIGN: Retrospective review in the setting of three tertiary care pediatric medical centers. METHODS: Methods included 1) extensive review of the literature; 2) a systematic review with respect to age, gender, presentation, associated medical problems, location and degree of subglottic narrowing, initial treatment, need for subsequent treatments, outcome, complications, and prognosis; and 3) statistical analysis to determine the effect of five variables (age, gender, degree of subglottic narrowing, location and extent of subglottic hemangioma, and lack or presence of other hemangioma) and the outcome of six different treatment modalities (conservative monitoring, corticosteroid, laser surgery, tracheotomy, laryngotracheoplasty, and interferon). RESULTS: In all, 116 patients with a mean age of 4.7 months were treated. The most common location of subglottic hemangioma was the left side. The range of subglottic narrowing was 10% to 99% (mean percentage, 65%). Twenty-six patients (22%) were managed with a single treatment modality, which included conservative monitoring (n = 13), corticosteroid (n = 11), and tracheotomy (n = 2). Ninety patients (78%) required multimodality treatments. Overall, the treatments included conservative monitoring (n = 13), corticosteroid (n = 100), tracheotomy (n = 32), CO2 laser (n = 66), interferon (n = 5), and laryngotracheoplasty (n = 25). Complication rates included the following: conservative monitoring (none), corticosteroid (18%), tracheotomy (none), CO2 laser (12%), interferon (20%), and laryngotracheoplasty (20%). The following variables showed statistical significance in the outcome of different treatment modality: 1) degree of subglottic narrowing (P < .001), 2) location of subglottic hemangioma (P < .01), and 3) presence of hemangioma in other areas (P < .005). Gender (P > .05) and age at the time of presentation (P > .06) did not show any statistical significance on the outcome of the treatments. CONCLUSION: Each patient should be assessed comprehensively, and treatment should be individualized based on symptoms, clinical findings, and experience of the surgeon. The authors presented treatment guidelines in an attempt to rationalize the management of subglottic hemangioma and to help determine the best possible treatment modality at the time of initial presentation.     

Open excision of subglottic hemangiomas to avoid tracheostomy

OBJECTIVE: To assess the efficacy of open excision as an alternative to tracheostomy in the management of subglottic hemangioma. DESIGN: A retrospective review of patients undergoing open surgical excision of subglottic hemangiomas over a 10-year period. SETTING: A tertiary pediatric center. PATIENTS: The study included 22 children ranging in age from 2 to 42 months (median age, 5 months) who underwent open excision of subglottic hemangioma over a 10-year period. RESULTS: Twenty-one patients were treated with single-stage procedures, with postoperative endotracheal intubation for an average of 5 days. One patient who had a preexisting tracheostomy was treated with a 2-stage procedure and underwent decannulation 2 months after excision. Seven other patients were tracheostomy dependent at the time of excision and underwent decannulation at the time of the procedure. Cartilage grafts were inserted in 10 patients. There were no problems with subglottic stenosis. Twenty-one patients reported good voice and no airway symptoms after a mean follow-up period of 42 months. Five patients had significant postoperative problems. Three patients required further endoscopic procedures for removal of granulation tissue, and 1 patient, who remains minimally symptomatic, developed an anterior glottic web. One patient required a 6-month course of steroids after surgery to treat residual glottic hemangioma. CONCLUSIONS: Open surgical excision of subglottic hemangiomas can be performed as a single procedure, avoiding a tracheostomy, when modern surgical techniques developed for laryngotracheal reconstruction are incorporated. This approach can avoid repeated endoscopic procedures, prolonged treatment with corticosteroids, and years spent with a tracheostomy waiting for spontaneous involution of the hemangioma.     

Surgical removal of subglottic hemangiomas in children.

OBJECTIVE: To examine the indications and the results of surgical excision of severe subglottic hemangiomas. DESIGN: Retrospective study and case series. SETTING: Four academic tertiary care centers of pediatric otolaryngology. PATIENTS: Twenty children were included from 1991 to 1997. All presented with severe subglottic hemangiomas resistant to classical treatments such as corticosteroids and/or CO2 laser. INTERVENTION: Six children were operated on using laryngotracheal reconstruction and prolonged stenting by a reinforced Silastic roll and 14 children were operated on by a single-stage laryngotracheoplasty and postoperative intubation in a pediatric intensive care unit. RESULTS: All patients were successfully decannulated or extubated and have been free from recurrent hemangiomas. CONCLUSION: The surgery of severe subglottic hemangiomas is a reliable technique in selected patients and should be considered in corticoresistant or corticodependent, circular, or bilateral hemangiomas.     

Subglottic hemangioma

Subglottic hemangioma is a rare condition that can be potentially life threatening because of airway obstruction. It is common for subglottic hemangioma to be misdiagnosed as croup initially. Infants with a subglottic hemangioma and cutaneous facial hemangiomas in a "beard" distribution should be evaluated for PHACE syndrome. Endoscopic laser resection is effective for subglottic hemangioma but carries a chance of subglottic stenosis, up to 25%. Open excision of subglottic hemangioma is an excellent option, particularly in patients with bilateral or circumferential subglottic hemangioma. It is a more extensive surgery when compared with endoscopic laser resection. Surgeons who do not have access to a pediatric intensive care unit staffed by experienced pediatric intensivists should not use this procedure.     

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.     

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.     

Systemic steroids for the management of obstructive subglottic hemangioma.

Subglottic hemangioma is a recognized cause of paediatric upper airway obstruction. We present 14 patients with subglottic hemangioma treated between 1984 and 1997,4 of whom had associated extralaryngeal hemangiomatous lesions (28%). The degree of upper airway obstruction ranged between 20% and 90%. Patients with subglottic hemangioma who had obstruction of the laryngeal lumen more than 25% and those with obstructive symptoms were treated with systemic steroids. The patients were followed clinically, radiographically, and with repetitive bronchoscopies. Nine of 10 patients (90%) have responded clinically to systemic steroids. There were no major complications from the systemic steroid treatment. One patient developed a cushingoid face that was reversed after the cessation of steroid therapy. The purpose of this study is to show that systemic steroids, with or without short-term intubation after diagnostic bronchoscopy, can be used as a safe and effective alternative in the management of obstructive paediatric subglottic hemangiomas.     

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.     

Adult laryngeal hemangioma CO2 laser excision. A single institution 3-year experience (Vittorio Veneto 2001-2003)

CONCLUSIONS: Adult supraglottic hemangiomas can be treated successfully with CO2 laser excision with limited morbidity. Extended laryngeal cavernous hemangiomas involving hypopharynx should be approached with staged CO2 laser surgical procedures. OBJECTIVES: Hemangioma of the adult larynx is an uncommon, benign lesion characterized by thin, friable mucosa overlying the vascular stroma. The optimal surgical approach to these lesions is still controversial because only anecdotal case reports or very limited series are available. We report a 3-year, retrospective, single institution study of the results of CO2 laser treatment of supraglottic hemangiomas in adults. PATIENTS AND METHODS: Six consecutive cases of adult laryngeal hemangioma were treated by the first author with CO2 laser microsurgery alone. RESULTS: In five of six cases, no recurrences have been diagnosed (median follow-up period: 29 months). One case presented limited persistence of disease in the retro-cricoid and arytenoids at 20-month follow-up control.     

The KTP laser: an emerging tool in pediatric otolaryngology

PURPOSE: To report the frequency and spectrum of use of the potassium titanyl phosphate (KTP) laser in a tertiary referral pediatric otolaryngology practice and to focus on a novel use for the KTP laser which has not previously been described in the literature. DESIGN: A retrospective chart review of the operative log database of a pediatric otolaryngologist in a tertiary referral setting over a seven year period. RESULTS: Out of 2886 cases, a total of 49 (1.7%) involved the use of the KTP laser. These included 7 otologic cases, 3 laryngeal cases, 31 subglottic/tracheal cases, 1 esophageal case and 7 nasal cases. One of these cases involved a previously unreported use of the KTP laser, closure of a tracheo-esophageal fistula (TEF). CONCLUSION: The KTP laser is an important operative tool in pediatric otolaryngology and new uses for this laser continue to emerge. One of these, KTP closure of a TEF offers pediatric otolaryngologists the potential for significant changes in management of this congenital problem with reduced surgical morbidity. Familiarity with the KTP laser and expertise in its use and applications is essential in providing state-of-the-art care to pediatric otolaryngology patients in a tertiary referral center.     

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.     

Propranolol for treatment of subglottic hemangioma

Subglottic hemangiomas are extremely dangerous due to its location and rapid growth during the proliferative phase. Many different treatments are described but these methods are still not satisfactory. Recently propranolol has been used as a new option in hemangioma therapy. We describe a case of 6-week infant with subglottic hemangioma discovered direct laryngoscopy, presented with dyspnoea and inspiratory stridor. After oral propranolol administration all baseline airway symptoms had resolved and endoscopic examination demonstrated significant regression of the hemangioma. We suggest that the propranolol should be used as a first-line treatment in subglottic hemangiomas in children.     

Management of infantile subglottic hemangioma: Acebutolol or propranolol?

The successful management of subglottic hemangioma with propranolol has been reported. We report three cases of subglottic hemangioma treated with the cardioselective beta-blocker acebutolol, 8 mg/kg/day. Treatment was efficient in two cases while an open procedure was necessary in the third child. In our experience, acebutolol could be easily administered in oral form twice-a-day only with a dose that was adaptable according to the growth of the child and showed no side effects.We also report a case of rebound growth after beta-mimetic drug use and the efficiency of propranolol treatment in such a recurrence.Considering the lack of side effects and the advantages in terms of administration, we suggest acebutolol as a first-line treatment of subglottic hemangiomas for which intervention is required.     

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.     

Management of subglottic hemangioma

PURPOSE OF REVIEW: Subglottic hemangioma is a challenging congenital anomaly involving the larynx in children. Management is not uniform, and no single treatment modality has been accepted as ideal. During the last several years only a few articles have dealt with this topic. The purpose of this review is to examine the published literature and discuss the alternative treatments of SGH. The authors also describe their approach and the use of microdebrider as a new surgical tool. RECENT FINDINGS: Observation is an option for the rare case of SGH with minimal airway obstruction. However, most cases will require some additional intervention. Systemic steroids intralesional steroid injection, laser ablation with both the CO2 and KTP lasers, interferon (IFN) and open surgical excision have all been utilized. SUMMARY: The authors found few technical innovations in this field in the last 5 years. All techniques utilized have some degree of success. However, all approaches have downsides and are associated with complications, some of which can be very serious. Ideally, more structured research comparing techniques would be helpful to best determine operative and postoperative management.     

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.     

Open surgical treatment of laryngo-tracheal stenoses in children

BACKGROUND: Treatment strategy in laryngo-tracheal stenoses in children has for a long time been conservative treatment with tracheostomy or bougination in hope of a more or less spontaneous resolution of the stenosis during growth of the child. The results of this option as well as the endoscopic treatment with different laser systems has proved to be rather disappointing. A child with a tracheostomy means a heavy load for the parents to look after this child as well as a constant threat from complications by displacement or plugging of the cannula. METHOD: A retrospective chart review of 22 children, aged between two months and 15 years at the time of surgery with laryngo-tracheal stenoses treated by different open surgical procedures. RESULTS: The aetiology of the stenoses was prolonged endotracheal intubation in 12 children, congenital stenoses in 3 children, unsuccessful laser treatment for acquired stenoses in 3 children, subglottic hemangioma in 3 children and a transglottic cyst in 1 child. 17 cases were treated by laryngo-tracheal reconstruction with rib cartilage graft, 3 crico-tracheal resections, and 2 laryngofissures. Five children could be treated without tracheostomy. From the remaining 17 cases 13 could be decannulated, 1 child died one week after surgery from his congenital heart disease. 3 children are still with a tracheostomy, two of them had had endoscopic laser therapy alio loco before. CONCLUSIONS: Open laryngo-tracheal surgery for paediatric airway stenoses is a successful treatment option besides endoscopic management for selected cases. Resection surgery seems to be indicated for severe stenoses with proliferative scar tissue formation. Depending on personal experience and post-operative facilities procedures without tracheostomy but prolonged post-operative intubation are possible single-stage-solutions.     

The use of propranolol in the treatment of subglottic hemangiomas: A literature review and meta-analysis

Objectives1) Describe the origins of the use of propranolol in the treatment of subglottic hemangiomas, 2) Perform meta-analysis of all case reports and series in which propranolol was used to treat subglottic hemangiomas.Study designLiterature review and meta-analysis.MethodsA total of 61 cases were identified from 19 scholarly articles. Cases were assessed by parameters including age at diagnosis, presence of other hemangiomas, percent airway obstructed, dose of propranolol, treatment duration, age at therapy termination, use of steroids, and treatment failure. Treatment failure was defined as: 1) Need for surgery after initiation of propranolol, 2) Return of symptoms, or 3) Endoscopic worsening/recurrence of hemangioma. All data was subjected to comprehensive statistical analysis.ResultsThough not statistically significant, a trend was noted towards a decreased treatment failure rate with increasing doses of propranolol (p = 0.0563). The use of concurrent steroids was associated with a higher failure rate (p = 0.0487). Notably, no associations were observed between the presence of additional hemangiomas, prior surgery, or increased initial percent airway obstruction with treatment failure.ConclusionPropranolol is rapidly becoming the standard of care in the treatment of subglottic hemangiomas. Despite widespread adoption, the rarity of this condition has limited previous studies to case reports and small series. No evidence-based guidelines exist for proper dosing of propranolol. The results of this meta-analysis suggest a benefit to higher doses of propranolol (3 mg/kg/day), though further investigation is needed.