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Abstract: Case Reports |

Supraglottic Hemangioma, Bronchoscopy Diagnostic of an Atypical Presentation of Laryngeal Stridor, Associated With Double Vascular Ring in an Infant FREE TO VIEW

Erick W. Bringas, Resident; G. Teresita Gutiérrez, MD; M. Jose Cota, MD; Daniel Carrasco, MD; M. Silvia Lule, MD
Author and Funding Information

INER, Mexico D.F., Mexico


Chest


Chest. 2003;124(4_MeetingAbstracts):324S-325S. doi:10.1378/chest.124.4_MeetingAbstracts.324S
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Abstract

INTRODUCTION:  Hemangioma represent only the 10% of the laryngeal pathology usually in the subglottic space, been the cavernous and capillary histologic types the most common, and the laryngeal stridor the mainly sing of clinic suspicion.We present an extremely rare case of Supraglottic hemangioma associated to extrinsic compression in a pediatric patient.

CASE PRESENTATION:  Male infant of eight month of age, without family history of cardiovascular disease, Delay on grown-up and ponderal deficit of 25%, with a history of laryngeal stridor since born, and remission of multiple exacerbation, until it became continuous; a diagnostic of laringotracheitis and recurrent neumonia was make, requiring hospitallary treatment in four times. Eigth days before admition present with biphasic stridor, without modification on postural changes, a treatment for laringotracheitis was stared for five days without respond and respiratory distress that required ventilatory support, and was send from a pediatric hospital to our institution (Instituto Nacional de Enfermedades Respiratorias of México D.F.) The Chest X ray showed complete intermitent left lung atelectasi and dynamic hyperinsuflation of contralateral lung.The whole bioquimical laboratory were in normal limits, arterial gas presented respiratory acidosis.A supraglottic exofitic red lesion of soft surface was present in bronchoscopy, just on the rigth ventricular band, occluding 90% of the hypofaring and the glottic and supraglottc lumen by a valvular efect, then was postulated the clinic suspection of hemangioma and extrinsic compression of the main rigth bronchus.The echocardiography reported moderate to severe pulmonary arterial hypertension with structural hart between normal limits; The helicoidal computed tomography suggested the posibility of a vascular ring, without the posibility of performed cardiac catheterization due to an a Streptococcus pyogenes bacteremia.A tracheotomy after lesión diagnostic was performed, with a satisfactory evolution weaning of mechanical ventilation around ten days; nevertheless he presented a sudden cardiorespiratory arrest due to plugging in the canul.The necropsy reported: supraglottic cavernous hemangioma and double vascular ring. Sling kind of the left pulmonary artery and right aortic arc.DISCUSSION: This is an unusual case presentation of supraglottic hemagioma due to the mayority of cases have a subglottic presentation and the association with a congenital vascular malformation of the left pulmonary artery and rigth aortic arc makes the case of interest due to the few cases reported in the literature and the posibility of a new multiple syndrome reported previously.CONCLUSIONS: The evaluation of stridor in pediatric population with bronchoscopy permit not only the valoration of the airway and to make some diferential diagnostic like foreing body, moreover it is an impotant diagnostic tool in this lesion that maybe evolve a special syndrome in this group of patient.

DISCLOSURE:  E.W. Bringas, None.

Wednesday, October 29, 2003

2:00 PM - 3:30 PM


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