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      CYSTIC HYGROMA CO-EXISTING WITH CONGENITAL SUBGLOTTIC STENOSIS

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          Abstract

          Respiratory distress is one of the commonest cause of admission into a Neonatal Intensive Care Unit, be it surgical or medical in nature. Adequate and prompt resuscitation as well as intubation can be life saving. Emergency or early tracheostomy may be necessary if airway intervention is needed. The authors present the case of a term neonate who was born with a large cervical cystic hygroma causing compression of the airway, together with a concurrent grade III subglottic stenosis, in respiratory distress within few minutes of life. Nine months post tracheostomy and sclero-therapy treatment twice, the child shows marked improvement. The succesful management of this unusual case of severe extrinsic compression with concurrent internal airway obstruction is presented.

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          Most cited references5

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          Congenital airway abnormalities in patients requiring hospitalization.

          To determine the cause of congenital airway abnormalities in pediatric patients requiring hospitalization for their respiratory status. Case series in a tertiary care center. A 5-year retrospective chart review was conducted at our institution. A total of 174 patients were identified who required hospitalization for their respiratory status as a result of a congenital airway abnormality. Of the 174 patients, 114 (65.5%) were male and 60 (34.5%) were female. Eighty patients (47%) presented within the first 3 months of life. Forty-six patients (26%) were born prematurely, and 49 patients (28%) were diagnosed as having gastroesophageal reflux. The majority of patients (139 [80%]) had multiple presenting symptoms or signs. Stridor was the most common (129 [74%]), followed by accessory respiratory effort, cyanosis, apnea, and failure to thrive. Diagnosis was made at the time of surgical evaluation in 91% of the patients, with the remaining diagnoses made using radiological findings and/or clinical evaluation. Sixty-five patients (37%) had multiple sites of airway abnormalities; laryngeal abnormalities were noted almost 3 times as often as tracheal abnormalities (161 vs 62, respectively). Of the laryngeal abnormalities, laryngomalacia was the most common, followed by glottic web, subglottic stenosis, vocal-cord paralysis, and subglottic hemangioma. Tracheomalacia was the most common tracheal abnormality, followed by external compression and tracheal stenosis. Thirty-three patients (19%) required tracheotomy for management of recurrent respiratory decompensation. While congenital airway abnormalities are usually self-limited, those patients requiring hospitalization represent a group with a more severe respiratory status who have a greater chance of requiring tracheotomy. The recognizable percentage of patients with gastroesophageal reflux and prematurity accounts for comorbid factors in the need for hospitalization for respiratory issues related to congenital airway abnormalities.
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            Embryology of the larynx

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              Cystic hygromas associated with Turner's syndrome. Report of three cases.

              Three cases of cystic hygromas are reported. Two of these were singleton pregnancies diagnosed at 20 weeks' gestation and the other was an affected fetus in a twin pregnancy at 19 weeks gestation. Amniocentesis was done and chromosomal analysis revealed Turner syndrome in all cases. The two singleton pregnancies were terminated by intrauterine installation of PgF2a. The affected fetus of the twin pregnancy died at 24 weeks' gestation and the pregnancy continued.
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                Author and article information

                Journal
                Acta Inform Med
                Acta Inform Med
                AIM
                Acta Informatica Medica
                AVICENA, d.o.o., Sarajevo
                0353-8109
                1986-5988
                September 2012
                : 20
                : 3
                : 192-193
                Affiliations
                Department of Otorhinolaryngology-Head & Neck Surgery, School of Medical Sciences, University Sains Malaysia, Kota Bharu, Kelantan, Malaysia.
                Author notes
                Corresponding author: Assoc Prof Baharudin Abdullah, MD, MBBS (Mal), MMED ORL-HNS (USM). Department of Otorhinolaryngology–Head and Neck Surgery, School of Medical Sciences, Universiti Sains Malaysia, Kubang Kerian 16150, Kelantan, Malaysia. Tel: +6097676416. Fax: +6097676424. E-mail: baharudin@ 123456kb.usm.my
                Article
                AIM-20-192
                10.5455/aim.2012.20.192-193
                3508856
                23322978
                4891a73f-fde0-44d9-bf54-9a56c6a42204
                © 2012 AVICENA

                This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License ( http://creativecommons.org/licenses/by-nc/3.0/) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.

                History
                : 3 June 2012
                : 13 July 2012
                Categories
                Case Report

                Bioinformatics & Computational biology
                cystic hygroma,respiratory distress.,sublottic stenosis

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