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Lipoid Proteinosis: A Rare Disease In Pediatric Dentistry.

Authors :
Lourenço AG
Araújo VC
Passador-Santos F
Sperandio M
Neville BW
Dorta RG
Source :
Brazilian dental journal [Braz Dent J] 2020 Mar-Apr; Vol. 31 (2), pp. 186-189.
Publication Year :
2020

Abstract

This report describes the diagnostic process of a rare disorder in a Brazilian female child. The patient presented initially as a 7-year-old with multiple whitish submucosal nodules of a fibrous consistency in the lower lip, but with an inconclusive pathology report. When she turned 9 years of age, she presented with exacerbation of the original clinical findings, which then involved the upper lip, buccal mucosa, tongue and lingual frenulum. In addition, dermatological lesions were noted on the child's limbs and face, as well as a hoarse voice. Histopathological examination of the buccal mucosa revealed dense connective tissue with hyaline foci, which were positive with periodic acid-Schiff (PAS) staining and resistant to diastase digestion. Clinical and histopathological findings led to the diagnosis of a rare genetic disease with fewer than 300 reported cases - lipoid proteinosis. Magnetic resonance imaging revealed calcium deposits in her amygdaloid region of the brain, and nasopharyngolaryngoscopy revealed lesions in her vocal cords. The patient currently is stable and under multidisciplinary follow-up, but no treatment has been recommended to date.

Details

Language :
English
ISSN :
1806-4760
Volume :
31
Issue :
2
Database :
MEDLINE
Journal :
Brazilian dental journal
Publication Type :
Academic Journal
Accession number :
32556019
Full Text :
https://doi.org/10.1590/0103-6440202003054