Propranolol for the treatment of airway hemangiomas: A case series and treatment algorithm

Objectives

(1) To present six patients with symptomatic airway hemangiomas treated with oral propranolol. (2) To review the diagnostic and treatment options for airway hemangiomas and propose a new management protocol.

Study design

Retrospective review.

Setting

Tertiary care children's hospital.

Subjects and methods

Pediatric patients diagnosed with obstructive airway hemangiomas treated with oral propranolol. Patients were followed for symptomatic improvement and relief of airway obstruction on imaging or laryngoscopy.

Results

Seven patients presenting with airway obstruction were treated with propranolol. One patient had a focal hemangioma confined to the subglottis. Four patients had airway hemangiomas that extended beyond the confines of the larynx and trachea. A sixth patient had a bulky supraglottic hemangioma. A seventh patient with an extensive maxillofacial lesion failed propranolol therapy and was found to have a pyogenic granuloma on final pathology after excision. Six patients had failed standard medical therapy and/or surgical interventions and were treated successfully with oral propranolol with improvements in airway symptoms and oral intake, requiring no further surgical intervention. Treatment was initiated as early as 1.5 months of age, and as late as 22 months. No adverse side effects of propranolol were noted.

Conclusions

Oral propranolol was successfully used to treat airway hemangiomas, resulting in rapid airway stabilization, obviating the need for operative intervention, and reducing the duration of systemic corticosteroid therapy while causing no obvious adverse effects. These outstanding results enable the possibility of use of a standardized diagnostic and treatment algorithm for airway hemangiomas that incorporates systemic propranolol.     

A meta-analysis on the effectiveness of propranolol for the treatment of infantile airway haemangiomas

Objective

To study the effectiveness of propranolol in infantile airway haemangiomas and compare the effectiveness of propranolol vs. different therapies.

Methods

A literature search of Ovid, Embase, the Cochrane database, Google™ Scholar, and Medline using PubMed as the search engine was performed to identify studies that analysed the effect of propranolol treatment in children with airway haemangiomas. Random-effect meta-analytical techniques were conducted for the outcome measures.

Results

Thirteen studies, comprising 36 patients were included in the analysis. Propranolol was found to be an effective intervention for the resolution of infantile airway haemangiomas (P < 0.00001). Meta-analysis of effectiveness of propranolol vs. steroids, CO₂ laser, or vincristine showed that propranolol is the most effective treatment.

Conclusions

This meta-analysis demonstrated that propranolol should be recommended as a first-line treatment in infantile airway haemangiomas. However, because of the possible side effects of propranolol, current infantile haemangioma treatment centres recommend a full cardiovascular and respiratory review be performed prior to initiation of therapy.     

Use of intravenous propranolol for control of a large cervicofacial hemangioma in a critically ill neonate

Cervicofacial segmental infantile hemangiomas (IH) may result in airway obstruction requiring use of propranolol to induce hemangioma regression and reestablish the airway. We present the first case using intravenous (IV) propranolol for control of airway obstruction and rapid expansion of cervicofacial IH in the setting of necrotizing enterocolitis (NEC) impaired gastrointestinal function. Intravenous dosing of propranolol was tolerated well in a critically ill neonate with multisystem complications of prematurity.     

Misdiagnosed as infantile hemangioma: Early presentation of small vessel-rich AVM

Arteriovenous malformations (AVMs) are typically present at birth but may not become clinically significant till later in life. The rarity of AVMs leads to misdiagnosis, as they can be confused in young patients with infantile hemangioma (IH). Small vessel-rich AVM can be radiologically consistent with an IH, while the clinical presentation is that of a slowly growing vascular anomaly. These cases highlight the clinical dilemma that while a vascular anomaly may appear by imaging studies to be the common and generally innocuous IH, an atypical growth pattern should raise clinical suspicion for an AVM.     

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.     

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。     

婴幼儿腮腺血管瘤的手术治疗

目的 :探讨治疗婴幼儿腮腺血管瘤的有效方法。方法 :19例婴幼儿腮腺血管瘤 ,行腮腺浅叶切除 15例 ,全腮腺切除 3例 ,腮腺浅叶切除加深叶瘤体缝扎术 1例。 6例皮肤受累均同期切除 ,3例拉拢缝合 ,3例缺损较大 ,行颈项皮瓣转移修复 ,术中采用“假包膜”外剥离法及顺行解剖面神经主干法。结果 :3例出现轻度面瘫 ,均在 3～ 6个月内恢复。术中仅 1例输血 2 0 0ml。所有病例随访 1年以上 ,无肿瘤复发。结论 :外科手术中采用“假包膜”外剥离法 ,顺行面神经解剖及受累皮肤切除后缺损行颈项皮瓣转移修复等治疗 ,是根治婴幼儿腮腺血管瘤的有效方法     

Initial experience using propranolol as the sole treatment for infantile airway hemangiomas

The objective of this study is to describe the initial use of propranolol as the sole treatment for focal infantile airway hemangiomas, and to report on available literature describing the use of propranolol for airway lesions. This retrospective case series was carried out at a tertiary pediatric medical center. We obtained the following results: two children demonstrated significant response to oral propranolol therapy and avoided not only invasive surgical procedures, but also long-term administration of oral corticosteroids. This is the first report of treating infantile airway hemangiomas with only propranolol without additional surgical intervention or corticosteroid use. Review of literature reveals initial case series with similar, successful results using propranolol as an adjuvant treatment along with other medications and surgical interventions. We conclude that the initial use of propranolol as the sole treatment for infantile airway hemangioma is promising. Literature review reveals that propranolol as the sole treatment for most head and neck hemangiomas shows significant promise based on early case reports. Further studies are needed to determine the long-term effectiveness, dosing strategies, and side effect profile of propranolol treatment for hemangiomas.     

Facial nerve hemangioma: A rare case involving the vertical segment

This case report and literature review reports on a rare case of facial nerve hemangioma (FNH) involving the vertical facial nerve (FN) segment, and discusses the clinical presentation, imaging, pathogenesis, and management of these rare lesions. A 53‐year‐old male presented with a 10‐year history of right hemifacial twitching and progressive facial paresis (House‐Brackmann grading score V/VI). The computed tomography and magnetic resonance imaging studies confirmed an expansile lesion along the vertical FN segment. Excision and histopathologic examination demonstrated FNH. FNHs involving the vertical FN segment are extremely rare. Despite being rare lesions, we believe that familiarity with the presentation and management of FNHs are imperative. Laryngoscope, 2012     

Postcricoid hemangioma presenting as dysphagia: a report of 4 cases

Hemangiomas involving the postcricoid region of the hypopharynx are rare. This report reviews our experience with 4 cases of postcricoid hemangioma, including a set of twin siblings. All patients underwent panendoscopy. All patients were managed nonsurgically. Three patients did well with dietary modification. One patient, with multiple medical problems, remains partially dependent on her gastrostomy tube. To our knowledge this report represents the largest series in the literature and the first to describe similar lesions in twin siblings. This report highlights the importance of a complete fiberoptic office examination of children who present with symptoms of dysphagia or aspiration.     

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.     

Intramuscular hemangioma of masseter muscle with prominent formation of phleboliths: a case report

A 14-year-old girl was referred for evaluation and management of progressive, painful swelling of the right cheek. Swelling had been present since 3-year old and had gradually increased in size. Magnetic resonance imaging (MRI) revealed a well-circumscribed, brighter mass in the right masseter muscle with numerous rounded areas of signal hypointensity. Preoperative diagnosis was intramuscular hemangioma of the masseter muscle and surgery was performed. The tumor was completely removed except for a few, small phleboliths. Pathological examination of resected tissue led to a high suspicion of cavernous hemangioma with phlebolithiasis. Nine months postoperatively, the patient developed another painful mass in the right masseter muscle. MRI indicated recurrent hemangioma and further surgery was performed. Careful exploration resulted in completely removal of residual phleboliths accompanied with fibroadipose tissue. Part of the buccal branch of the facial nerve was excised to achieve complete resection of the lesion. Histological examination revealed distinct venous formation in phleboliths adjacent to fibroadipose tissue, demonstrating that both phleboliths and feeding vessels had been left by the previous operation. The present report reviews the literature on intramuscular hemangiomas of the masseter muscle, and discusses diagnostic methods and optimal surgical treatment.     

Propranolol in the therapeutic strategy of infantile laryngotracheal hemangioma: A preliminary retrospective study of French experience

Objective

Preliminary assessment of the efficacy of propranolol on subglottic hemangioma in children on a nation-wide scale.

Methods

Multicentric, retrospective study of clinical files of 14 children; pre- and post-treatment endoscopies.

Results

Mean age at diagnosis was 2.3 (0.7-4) months. Mean percentage of airway obstruction was 68% (15-90) before propranolol introduction. Propranolol was started at 5.2 (0.7-16) months of age. This treatment was effective in all cases with a mean regression of the stenosis to 22% after 2 weeks and 12% after 4 weeks. Other medical treatments (steroids) could be stopped. In one patient, a side effect of propranolol motivated the switch to another β-blocker. In four patients, treatment was stopped after 5.2 (1-10) months with a relapse in 2 (50%) cases. One of these two patients developed a resistance to propranolol and required a surgical procedure by external approach.

Conclusion

This preliminary nation-wide survey confirms propranolol high effectiveness against airways’ localization of infantile hemangiomas. Propranolol also allows alleviation or cut-off of previous medical treatments. However, recurrences are possible after early treatment interruption.     

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.