Gastrointestinal Involvement in Lipoid Proteinosis: A Ten-Year Follow-Up of a Brazilian Female Patient

Lipoid proteinosis is a rare autosomal recessive disease characterized by the deposition of hyaline material in the skin and internal organs. The main clinical features are hoarseness and typical skin lesions. In this report we describe the endoscopic and radiologic findings in a Brazilian female pa...

Full description

Saved in:
Bibliographic Details
Main Authors: Juliana Custódio Lima, Cristiane Kibune Nagasako, Ciro Garcia Montes, Irene Harumi Kamata Barcelos, Rita Barbosa de Carvalho, Maria Aparecida Mesquita
Format: Article
Language:English
Published: Wiley 2014-01-01
Series:Case Reports in Medicine
Online Access:http://dx.doi.org/10.1155/2014/952038
Tags: Add Tag
No Tags, Be the first to tag this record!
_version_ 1850236139385913344
author Juliana Custódio Lima
Cristiane Kibune Nagasako
Ciro Garcia Montes
Irene Harumi Kamata Barcelos
Rita Barbosa de Carvalho
Maria Aparecida Mesquita
author_facet Juliana Custódio Lima
Cristiane Kibune Nagasako
Ciro Garcia Montes
Irene Harumi Kamata Barcelos
Rita Barbosa de Carvalho
Maria Aparecida Mesquita
author_sort Juliana Custódio Lima
collection DOAJ
description Lipoid proteinosis is a rare autosomal recessive disease characterized by the deposition of hyaline material in the skin and internal organs. The main clinical features are hoarseness and typical skin lesions. In this report we describe the endoscopic and radiologic findings in a Brazilian female patient presenting extensive gastrointestinal involvement and the evolution of the detected lesions in ten years of follow-up. Initial upper endoscopy and colonoscopy showed a similar pattern of multiple yellowish nodules throughout the esophagus, stomach, duodenum, and colons. Histological analysis confirmed the diagnosis of lipoid proteinosis. In addition, small bowel follow through demonstrated numerous well defined, round, small filling defects throughout the jejunum. Ten years later, the esophageal lesions remained the same, but none of the previous alterations were detected in the stomach, duodenum, and colons. In conclusion, lipoid proteinosis may affect all gastrointestinal organs with the same pattern of macroscopic and microscopic lesions. Some lesions may regress with increasing age.
format Article
id doaj-art-1546760e5f044078880717fd4ea54964
institution OA Journals
issn 1687-9627
1687-9635
language English
publishDate 2014-01-01
publisher Wiley
record_format Article
series Case Reports in Medicine
spelling doaj-art-1546760e5f044078880717fd4ea549642025-08-20T02:02:02ZengWileyCase Reports in Medicine1687-96271687-96352014-01-01201410.1155/2014/952038952038Gastrointestinal Involvement in Lipoid Proteinosis: A Ten-Year Follow-Up of a Brazilian Female PatientJuliana Custódio Lima0Cristiane Kibune Nagasako1Ciro Garcia Montes2Irene Harumi Kamata Barcelos3Rita Barbosa de Carvalho4Maria Aparecida Mesquita5Gastroenterology Unit, Faculty of Medical Sciences, State University of Campinas (UNICAMP), 13083-887 Campinas, SP, BrazilGastrocenter, State University of Campinas (UNICAMP), 13083-878 Campinas, SP, BrazilGastroenterology Unit, Faculty of Medical Sciences, State University of Campinas (UNICAMP), 13083-887 Campinas, SP, BrazilDepartment of Radiology, Faculty of Medical Sciences, State University of Campinas (UNICAMP), 13083-887 Campinas, SP, BrazilGastrocenter, State University of Campinas (UNICAMP), 13083-878 Campinas, SP, BrazilGastroenterology Unit, Faculty of Medical Sciences, State University of Campinas (UNICAMP), 13083-887 Campinas, SP, BrazilLipoid proteinosis is a rare autosomal recessive disease characterized by the deposition of hyaline material in the skin and internal organs. The main clinical features are hoarseness and typical skin lesions. In this report we describe the endoscopic and radiologic findings in a Brazilian female patient presenting extensive gastrointestinal involvement and the evolution of the detected lesions in ten years of follow-up. Initial upper endoscopy and colonoscopy showed a similar pattern of multiple yellowish nodules throughout the esophagus, stomach, duodenum, and colons. Histological analysis confirmed the diagnosis of lipoid proteinosis. In addition, small bowel follow through demonstrated numerous well defined, round, small filling defects throughout the jejunum. Ten years later, the esophageal lesions remained the same, but none of the previous alterations were detected in the stomach, duodenum, and colons. In conclusion, lipoid proteinosis may affect all gastrointestinal organs with the same pattern of macroscopic and microscopic lesions. Some lesions may regress with increasing age.http://dx.doi.org/10.1155/2014/952038
spellingShingle Juliana Custódio Lima
Cristiane Kibune Nagasako
Ciro Garcia Montes
Irene Harumi Kamata Barcelos
Rita Barbosa de Carvalho
Maria Aparecida Mesquita
Gastrointestinal Involvement in Lipoid Proteinosis: A Ten-Year Follow-Up of a Brazilian Female Patient
Case Reports in Medicine
title Gastrointestinal Involvement in Lipoid Proteinosis: A Ten-Year Follow-Up of a Brazilian Female Patient
title_full Gastrointestinal Involvement in Lipoid Proteinosis: A Ten-Year Follow-Up of a Brazilian Female Patient
title_fullStr Gastrointestinal Involvement in Lipoid Proteinosis: A Ten-Year Follow-Up of a Brazilian Female Patient
title_full_unstemmed Gastrointestinal Involvement in Lipoid Proteinosis: A Ten-Year Follow-Up of a Brazilian Female Patient
title_short Gastrointestinal Involvement in Lipoid Proteinosis: A Ten-Year Follow-Up of a Brazilian Female Patient
title_sort gastrointestinal involvement in lipoid proteinosis a ten year follow up of a brazilian female patient
url http://dx.doi.org/10.1155/2014/952038
work_keys_str_mv AT julianacustodiolima gastrointestinalinvolvementinlipoidproteinosisatenyearfollowupofabrazilianfemalepatient
AT cristianekibunenagasako gastrointestinalinvolvementinlipoidproteinosisatenyearfollowupofabrazilianfemalepatient
AT cirogarciamontes gastrointestinalinvolvementinlipoidproteinosisatenyearfollowupofabrazilianfemalepatient
AT ireneharumikamatabarcelos gastrointestinalinvolvementinlipoidproteinosisatenyearfollowupofabrazilianfemalepatient
AT ritabarbosadecarvalho gastrointestinalinvolvementinlipoidproteinosisatenyearfollowupofabrazilianfemalepatient
AT mariaaparecidamesquita gastrointestinalinvolvementinlipoidproteinosisatenyearfollowupofabrazilianfemalepatient