Infantile hemangioma is the most common benign tumor of infancy, affecting 1–2% of infants. Hemangiomas of the airway constitute an even smaller percentage, but their management can be challenging due to the potential for life threatening airway compromise. A Subglottic hemangioma (SGH) makes up 1.5% of all congenital laryngeal anomalies, it is twice more common in females than males and have been linked to low birth weight and prematurity. It is during the early proliferative phase (1–3 months of life) that patients became symptomatic, developing characteristic stridor which may progress to respiratory distress. In this early stage, a SGH is often mistaken for a more common condition such as croup. The aim of this case is to underline what a recurrent dyspnea or laryngeal stridor in the first 6 months might hide Case report A 4 months-old-girl who was born at term of natural childbirth (birth weight 2.500 kg-SGA), presented with several weeks of unremitting stridor, substernal retractions. She was diagnosed to have bronchiolitis and she had been hospitalized twice in an another hospital and treated with oral steroids and nebulized racemic epinephrine without significant improvement in her symptoms; Than she had been sent to our hospital. She had intercostal and subcostal retractions. Both lungs had equal contribution to respiration, respiratory sounds were coarse and she had both inspiratory and expiratory stridors which were more obvious on bilateral sibilant rales, and inspiratory phase. She also had wheezing. Laboratory tests, echocardiogram and electrocardiogram were normal. Her follow-up showed that she was not responding to treatment and her respiratory distress was increasing, thus she had a laryngofibroscopy that did not reveal any clear structural abnormalities, and performed a CT scan of the neck, that revealed a laryngeal mass, confirmed by an MRI of the neck, which showed a solid tissues of low intensity on T1-weighted spin-echo images and of hyperintensity on T2-weighted spinecho images (6x8 mm), compatible with SGH. After her workup was complete, she received an initial dose of propranolol at 0.5 mg/kg, which was increased to 2 mg/kg, and no adverse effects were noted. SGH is a rare but potentially lifethreatening disease. A high index of suspicion is vital for the early, accurate diagnosis of this disease. Propranolol treatment has many advantages, it is non-invasive and it has a low complication rate; thus, the use of propranolol as a first-line treatment for SGH is proposed

A CASE OF A STRANGE DYSPNEA

CORETTI, ANTONELLA;Teresa Merola;Jessica Cinalski;Francesca Lotti;LANZANO, ANNA;DI FLORIO, LAURA;Valeria Verrotti di Pianella;Agostino Petraccaro;Enrica Manca;Rossella Giorgio;Sofia Siena;Isabella Patisso;Loredana Dipasquale;Michaela Sciacovelli;Annarita Pedico;Antonio Marseglia;Massimo Pettoello Mantovani.
Conceptualization
2019-01-01

Abstract

Infantile hemangioma is the most common benign tumor of infancy, affecting 1–2% of infants. Hemangiomas of the airway constitute an even smaller percentage, but their management can be challenging due to the potential for life threatening airway compromise. A Subglottic hemangioma (SGH) makes up 1.5% of all congenital laryngeal anomalies, it is twice more common in females than males and have been linked to low birth weight and prematurity. It is during the early proliferative phase (1–3 months of life) that patients became symptomatic, developing characteristic stridor which may progress to respiratory distress. In this early stage, a SGH is often mistaken for a more common condition such as croup. The aim of this case is to underline what a recurrent dyspnea or laryngeal stridor in the first 6 months might hide Case report A 4 months-old-girl who was born at term of natural childbirth (birth weight 2.500 kg-SGA), presented with several weeks of unremitting stridor, substernal retractions. She was diagnosed to have bronchiolitis and she had been hospitalized twice in an another hospital and treated with oral steroids and nebulized racemic epinephrine without significant improvement in her symptoms; Than she had been sent to our hospital. She had intercostal and subcostal retractions. Both lungs had equal contribution to respiration, respiratory sounds were coarse and she had both inspiratory and expiratory stridors which were more obvious on bilateral sibilant rales, and inspiratory phase. She also had wheezing. Laboratory tests, echocardiogram and electrocardiogram were normal. Her follow-up showed that she was not responding to treatment and her respiratory distress was increasing, thus she had a laryngofibroscopy that did not reveal any clear structural abnormalities, and performed a CT scan of the neck, that revealed a laryngeal mass, confirmed by an MRI of the neck, which showed a solid tissues of low intensity on T1-weighted spin-echo images and of hyperintensity on T2-weighted spinecho images (6x8 mm), compatible with SGH. After her workup was complete, she received an initial dose of propranolol at 0.5 mg/kg, which was increased to 2 mg/kg, and no adverse effects were noted. SGH is a rare but potentially lifethreatening disease. A high index of suspicion is vital for the early, accurate diagnosis of this disease. Propranolol treatment has many advantages, it is non-invasive and it has a low complication rate; thus, the use of propranolol as a first-line treatment for SGH is proposed
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Utilizza questo identificativo per citare o creare un link a questo documento: https://hdl.handle.net/11369/379291
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