Congenital nasal pyriform aperture stenosis: report of two cases
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CASE REPORT
Congenital nasal pyriform aperture stenosis: report of two cases Rafael Ferreira Zatz 1 & Julia Alice Cavicchioli Goldenberg 2 Dov Charles Goldenberg 1
&
Vânia Kharmandayan 1
&
Rolf Gemperli 1
&
Received: 26 June 2020 / Accepted: 12 August 2020 # Springer-Verlag GmbH Germany, part of Springer Nature 2020
Abstract Congenital nasal pyriform aperture stenosis is a rare cause for congenital nasal obstruction. There are few publications about this severe condition. We report two cases of newborns diagnosed with congenital nasal pyriform aperture stenosis submitted to surgical treatment. Both had respiratory difficulty since birth, represented clinically by the difficulty during breastfeeding. No concomitant comorbidities were identified. The first patient underwent early surgical treatment, as she presented with severe respiratory distress. The second patient had failed conservative treatment and was also subjected to the surgical widening of the pyriform aperture. Both patients evolved uneventfully in the postoperative period. Congenital nasal pyriform aperture stenosis is part of the differential diagnosis of congenital nasal obstruction, being choanal atresia the most common finding. Its clinical presentation can vary from mild respiratory distress, which only disables breastfeeding, to respiratory failure at rest. As it is a rare occurrence, with few publications, its management is still controversial. The need for surgical management in both reported cases and a favorable evolution are highlighted, allowing hospital discharge and adequate breastfeeding. Level of evidence: Level V, therapeutic study. Keywords Nasal obstruction . Congenital . Pyriform aperture stenosis . Neonates . Upper airway . Obstruction
Introduction Congenital nasal obstruction occurs in 1 in every 5000 neonates. It manifests at birth or in the first days of life through upper airway obstruction. The discomfort can be mild, and difficulty in breastfeeding is observed. When more severe, it may require admission to a Nasal Intensive Care Unit and even may need definitive airway access. In newborns, the most common diagnosis is bilateral choanal atresia. A possible differential diagnosis is congenital nasal pyriform aperture stenosis, which, despite being severe, is an entity little reported in the literature [1,2]. Difficulty or inability of passing a probe through the nostril sill alerts to this diagnosis.
* Dov Charles Goldenberg [email protected] 1
Division of Plastic Surgery, Hospital das Clínicas da Faculdade de Medicina da Universidade de São Paulo, Av. Dr. Enéas de Carvalho Aguiar, 255, Cerqueira César, São Paulo, Brazil
2
Faculdade de Medicina da Pontifícia Universidade Católica de São Paulo, Sorocaba, Brazil
Congenital nasal pyriform aperture stenosis is a rare cause of neonatal nasal obstruction. It is caused by the narrowing of the pyriform aperture due to dysplasia in bone formation of the nasal maxillary process. The reduced width leads to increased nasal airway resistance and upper airway obstructi
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