Case report
Role of Propranolol in the therapeutic strategy of infantile laryngotracheal hemangioma

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Abstract

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.

Introduction

Subglottic and/or tracheal infantile hemangiomas are potentially life-threatening tumors, despite their spontaneous regression typically beginning after 18–24 months of age. Many types of treatment have been proposed, some of them aiming to wait for spontaneous regression (tracheotomy), and some aiming to partially or totally reduce or destroy the hemangioma (corticosteroids, Interferon or Vincristine therapy, LASER, cryotherapy, local steroid therapy with intubation, open surgery) [1], [2], [3], [4], [5].

The adverse effects of these various types of therapies are potentially severe. Tracheotomy in infants has a high mortality/severe morbidity rate, 1–3% [6]. Long-term steroid therapy induces Cushing syndrome with growth retardation, typical appearance, hirsutism, arterial hypertension, hypertrophic cardiomyopathy, delayed wound healing, immunosuppression, and an infectious risk [7]. Interferon α2A and 2B are associated with many various side effects (flu type malaises, spastic diplegia, neutropenia, liver enzymes abnormalities) [8], [9], [10]. Vincristine therapy has been described in extensive life-threatening cases [1]. Side effects such as constipation, abdominal pain, and parasthaesia due to the peripheral neuropathy have been reported. Although there is resolution of symptoms after cessation of treatment, this last neurological side effect may impose a premature arrest of therapy. Local injections of steroids associated with intubation leads to prolonged hospitalization on the intensive care unit (ICU) (mean duration 19 days [3]). The main side effect of cryotherapy and LASER is pathological scarring with secondary laryngeal stenosis [11] and many teams only use LASER in small unilateral hemangiomas to limit this risk. The use of open surgery in bilateral or circumferential hemangiomas offers an effective therapeutic strategy. However, there is a risk of severe complications with this surgery, as in all open airway surgery.

The spectacular effect of Propranolol therapy on hemangiomas, described for the first time in 2008 by Leaute-Labreze [12] is dramatically changing the therapeutic strategies used to date.

Here we report the cases of two children presenting severe subglottic hemangioma successfully treated with Propranolol.

This study received the approval of the Institutional Review Board of our hospital. Informed consent for publication was obtained from the parents of both children.

Section snippets

Case No. 1

A 2-month-old girl was referred to our department for a PHACES syndrome with upper airway obstruction. The PHACES syndrome associates posterior fossa malformations, hemangiomas, arterial anomalies, coarctation of the aorta and other cardiac defects, eye abnormalities, and sternal defects. Usually, a single structural anomaly is associated with hemangiomata.

The child had a large sternal cleft (Fig. 1) diagnosed by antenatal ultrasonography, and a supra umbilical median raphe, which had partly

Discussion

Due to their variation of size, shape, and behavior, subglottic and/or tracheal infantile hemangiomas in children may present with a broad spectrum of clinical features, from a well-tolerated non-symptomatic tumor to a life-threatening lesion. Long-term complications, like increased pulmonary arterial pressure, may also occur in children with a chronic upper airway obstruction [14].

The use of tracheotomy in symptomatic lesions has been proposed as a “wait-and-see” treatment, to wait for the

References (17)

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