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.     

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.     

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.     

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

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.     

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.     

Treatment of infantile subglottic hemangioma. A report of 49 cases

Forty-nine laryngeal subglottic hemangiomas were observed in infants over a 9-year period. The diagnosis was always confirmed by direct laryngoscopy. The outcome of 42 of these hemangiomas was studied. A total of 26 infants recovered completely after medical treatment and 16 were intubated; only 4 underwent a tracheostomy. Short-term steroid therapy was sufficient in 7 infants, but long-term administration of corticosteroids was necessary in 14 cases. In 10 of the 16 intubated cases it was possible to withdraw corticosteroids permanently. Of the 6 remaining cases, 32P contact radiation therapy was tried in 2 cases of which one was successful, CO2 laser in 2 cases without success, tracheostomy alone in one case and one child died 2 months after extubation, without dyspnea. This series leads us to recommend corticosteroids as a first therapeutic step and short-term nasotracheal intubation if unsuccessful.     

Subglottic hemangiomas in infants: Treatment with CO2 laser

Subglottic hemangiomas in infants are rare but potentially lethal. Although the majority tend to regress after 12 to 18 months, lethal compromise of the airway is always possible until then, A plethora of treatments have been advocated for subglottic hemangiomas, each with significant morbidity. Eleven consectuve patients have undergone laser resection of subglottic hemangiomas at the University of California, San Francisco. Three infants have been managed without a tracheotomy. If a tracheotomy is required, earlier removal can be achieved with laser resection. No subglottic stenosis or hemorrhage has occurred following laser resection. CO₂ laser resection is now the safest and most effective treatment available.     

Superselective embolization of laryngeal hemangioma

Diffuse upper respiratory hemangiomas are extremely rare lesions. Unlike the much more common congenital subglottic hemangiomas, diffuse upper respiratory hemangiomas do not resolve spontaneously and may prove quite recalcitrant to conventional forms of therapy. Superselective embolization, a highly successful technique for the management of head and neck vascular lesions, was used to effectively control an obstinate laryngotracheal hemangioma. Superselective embolization appears to provide yet another viable therapeutic option in the management of laryngeal hemangiomas.     

Role of Propranolol in the therapeutic strategy of infantile laryngotracheal hemangioma

There are recent reports of effective treatment of cutaneous hemangiomas with Propranolol. The current study aims to assess efficacy of systemic Propranolol for subglottic hemangiomas and to discuss its place among the other available therapies. We report 2 infants with subglottic hemangiomas, which were resistant to other established medical treatments. One infant presented with PHACES association, the other with widespread cutaneous congenital hemangiomas. Both were subsequently treated with systemic Propranolol. Both patients’ subglottic hemangiomas responded dramatically to systemic Propranolol. No side effects of the therapy occurred, and a safety protocol previously designed for Propranolol prescribed for other indications was applied to our patients. Propranolol appears to be an effective treatment for subglottic hemangiomas and should now be used as a first-line treatment in subglottic hemangiomas when intervention is required.     

Infantile supraglottic hemangioma: a case report

Hemangiomas of the airway are benign vascular lesions that can involve any site from the nares to the tracheobronchial tree. Most of these lesions are seen in the subglottic area in infants. Supraglottic infantile hemangiomas are very rare. We report a case of supraglottic hemangioma in a 2-month-old boy who had been admitted to our hospital with inspiratory stridor and dyspnea. The hemangioma involved the left arytenoid and aryepiglottic fold. A tracheostomy was performed, and the patient was followed up endoscopically every 6 months thereafter The hemangioma disappeared when the child was 30 months old. Subsequently, a Montgomery T-tube was placed for 6 months to assist in maintaining normal breathing. The patient remains disease-free during ongoing follow-up. We also discuss the management strategies for infantile laryngeal hemangiomas.     

The treatment of subglottic hemangiomas of infants with prednisone

Because of their spontaneous regression, capillary and cavernous hemangiomas of infants usually do not require treatment. However, when they interfere because of their location with important functions of the body or even threaten life, treatment becomes mandatory. This is the case in most patients with congenital subglottic hemangiomas. In the past various methods of treatment have been used. All of them have disadvantages, and some are hazardous. Most cases (74.2%) required protracted tracheostomy. Mortality rate is still high (23.8%).We report herewith 5 infants with subglottic hemangiomas successfully treated with prednisone. No remarkable side-effects were observed, besides transient moonfacing. Similar good results were previously reported in 9 cases by several authors. Prednisone therapy if instituted early may reduce the need of tracheostomy and bring about complete recovery in this sometimes severe condition. Proper dosage and sufficient length of treatment are absolutely essential for such successful outcome.     

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.     

Case report: Treatment failure using propanolol for treatment of focal subglottic hemangioma

Subglottic hemangioma is a rare, potentially life threatening tumor of infancy which poses serious treatment challenges. A number of medical and surgical therapies over the years have met with variable success, and are associated with numerous potential morbidities. A potential windfall in the management of infantile hemangiomas has arisen with the recent identification of propanolol as a highly efficacious and relatively safe new treatment modality. At least five reports in the literature have described the rapid, successful treatment of airway hemangiomas with oral propanolol. We describe the first reported treatment failure with propanolol for subglottic hemangioma in an infant who initially responded dramatically to the medication.     

The open surgical approach to subglottic hemangioma

The management of subglottic hemangioma in infants remains controversial. We report two patients in whom an open surgical approach was utilized to completely excise the subglottic hemangioma. Both children have been successfully extubated and have remained symptom-free since that time.     

Management of middle ear hemangiomas

Hemangioma rarely presents as an isolated middle ear lesion. Because congenital hemangiomas usually regress spontaneously, surgical excision is not always necessary. However, a hemangioma in the middle ear can be complicated by infection and hearing impairment. We present 2 cases to show contrasting management strategies, both with successful outcomes. Two children who presented with unilateral otitis media were found to have concomitant mesotympanic hemangiomas on examination. The first child was asymptomatic and subsequently had an incisional biopsy, confirming the suspected diagnosis. The residual tumor then involuted over the following year. The second child, however, developed chronic otitis media refractory to medical therapy and required surgical removal of the entire hemangioma. Once a tissue diagnosis is made, middle ear hemangiomas can be managed expectantly (ie, wait for spontaneous resolution) or surgically. If growth of a middle ear hemangioma appears to be causing complications refractory to conservative therapy, then early surgical excision may be indicated.     

Head and neck hemangiomas of infancy

PURPOSE OF REVIEW: Hemangiomas are the most common benign tumor in infancy, affecting approximately 10% of infants. More than half of hemangiomas involve the head and neck. Increased understanding of hemangiomas has come about from identification of immunohistochemical markers, developmental defects associated with certain hemangiomas, and morphologic and classification schemes (focal versus segmental). RECENT FINDINGS: Immunohistochemical markers have been identified which are specific to hemangiomas in all phases of development and involution. Morphologic subtypes and anatomic locations have been identified that place an infant at higher risk for complications from the hemangioma. Hemangiomas associated with other developmental anomalies have been identified, which help guide the treating physician to tease out which infants will need more complete systemic investigations or imaging. Importantly for surgeons, studies have continued to identify which lesions may benefit from early intervention, either surgical or medical. SUMMARY: While full understanding of the mechanisms that turn on and turn off hemangiomas of infancy is not complete, progress has been made in identification of markers, subtypes at increased risk for complications, and in treatment. With continued work in these areas, we have increased knowledge of treatment options, optimal timing of surgical intervention, and ultimately, preventive options.     

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.     

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.     

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。