Case report
Management of infantile subglottic hemangioma: Acebutolol or propranolol?

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Abstract

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.

Introduction

Congenital subglottic hemangioma is the most common neoplasm of the airway in children and is potentially life-threatening during the proliferative phase (occurring below the age of 6–12 months), necessitating medical or surgical intervention. This may consist of a tracheotomy, general or local treatment with steroids, interferon or vincristine therapy, LASER treatment, or open surgery with laryngo-tracheal reconstruction, all of which carry a risk of adverse effects. Spontaneous regression typically occurs after 18–24 months of age.

Recent reports have been made of the effective treatment of cutaneous [1], [2], [3] and subglottic hemangiomas [4], [5], [6] with propranolol, a non-cardioselective beta-adrenoceptor blocking agent. This drug does however carry some potential side effects including bronchoconstriction, bradycardia, hypotension, and reduced physiological responses to hypoglycemia, as well as the drawback of needing administering three times daily. Another beta-adrenoceptor blocking agent, acebutolol, is not only cardioselective, but possesses both partial agonist (intrinsic sympathomimetic) and membrane stabilizing activity, can be administered twice daily, and has significantly fewer side effects on resting heart rate than propranolol, metoprolol and atenolol [7]. It seems therefore that acebutolol may prove more useful in treating subglottic hemangiomas.

Here we report our experience in four infants of treating subglottic hemangioma with either propranolol or acebutolol.

Section snippets

Clinical presentation

Four female infants were referred to our department for respiratory distress relating to a subglottic hemangioma with a laryngeal stenosis ranging from 70 to 90% with microlaryngoscopy and bronchoscopy (MLB) examination. Age at presentation ranged from 1.5 to 3 months. The subglottic hemangioma was associated with cutaneous hemangioma in cases 2 and 4 (neither had PHACES syndrome). Case 4 presented with an interventricular septal defect with right ventricular stenosis. The location of each

Discussion

In order to prevent potentially life-threatening complications associated with subglottic hemangioma during the proliferative phase (below the age of 6 months), medical or surgical intervention is necessary. Nevertheless, all treatment options to date themselves expose the child to potentially severe complications. Recently, the use of propranolol or acebutolol has been described in children with cutaneous hemangiomas [1], [2], [3] with success in almost all cases.

In this paper, we report three

Conclusion

Treatment of subglottic hemangiomas with acebutolol was efficient at controlling hemangioma volume. It was well tolerated in our patients with no reported side effects, and was easier to administer compared to propanolol. Once diagnosis has been confirmed, we propose the immediate initiation of acebutolol. The equal efficiencies of acebutolol and propanolol need to be demonstrated in a randomized controlled study which we are now working towards. Steroids should be used only in cases of

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