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Juvenile diabetes and systemic sclerosis: just a coincidence?
BACKGROUND: Limited joint mobility (LJM), previously known as cheiroarthropathy, refers to the presence of reduced extension at the finger joints in people with diabetes and may be associated with scleroderma-like syndromes such as diabetic sclerodactyly. While scleroderma-like syndromes and LJM hav...
Autores principales: | , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
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BioMed Central
2022
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Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9465903/ https://www.ncbi.nlm.nih.gov/pubmed/36089600 http://dx.doi.org/10.1186/s12969-022-00741-3 |
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author | Mastrangelo, Greta Meneghel, Alessandra Martini, Giorgia Moretti, Carlo Zulian, Francesco |
author_facet | Mastrangelo, Greta Meneghel, Alessandra Martini, Giorgia Moretti, Carlo Zulian, Francesco |
author_sort | Mastrangelo, Greta |
collection | PubMed |
description | BACKGROUND: Limited joint mobility (LJM), previously known as cheiroarthropathy, refers to the presence of reduced extension at the finger joints in people with diabetes and may be associated with scleroderma-like syndromes such as diabetic sclerodactyly. While scleroderma-like syndromes and LJM have been observed in patients with long-term diabetes and associated complications, the coexistence of diabetes with Juvenile systemic sclerosis (jSSc) is rarely described. CASE PRESENTATION: We describe the case of a 14-year-old boy with long-lasting type 1 diabetes (T1D) and suspected LJM associated with Raynaud phenomenon, sclerodactyly and tapering of the fingertips. A comprehensive work-up showed positive autoantibodies (ANA, anti-Ro-52, anti-Mi-2b), abnormal nailfold capillaroscopy with a scleroderma pattern, interstitial lung disease and cardiac involvement. The overall clinical picture was consistent with the diagnosis of jSSc. CONCLUSIONS: LJM can be the initial sign of underlying systemic sclerosis. Nailfold capillaroscopy may help differentiate jSSc from classical LJM in pediatric patients with T1D and finger contractures or skin induration of no clear origin. This case report provides a starting point for a novel hypothesis regarding the pathogenesis of jSSc. The association between T1D and jSSc may be more than a coincidence and could suggest a relationship between glucose metabolism, fibrosis and microangiopathy. |
format | Online Article Text |
id | pubmed-9465903 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2022 |
publisher | BioMed Central |
record_format | MEDLINE/PubMed |
spelling | pubmed-94659032022-09-13 Juvenile diabetes and systemic sclerosis: just a coincidence? Mastrangelo, Greta Meneghel, Alessandra Martini, Giorgia Moretti, Carlo Zulian, Francesco Pediatr Rheumatol Online J Case Report BACKGROUND: Limited joint mobility (LJM), previously known as cheiroarthropathy, refers to the presence of reduced extension at the finger joints in people with diabetes and may be associated with scleroderma-like syndromes such as diabetic sclerodactyly. While scleroderma-like syndromes and LJM have been observed in patients with long-term diabetes and associated complications, the coexistence of diabetes with Juvenile systemic sclerosis (jSSc) is rarely described. CASE PRESENTATION: We describe the case of a 14-year-old boy with long-lasting type 1 diabetes (T1D) and suspected LJM associated with Raynaud phenomenon, sclerodactyly and tapering of the fingertips. A comprehensive work-up showed positive autoantibodies (ANA, anti-Ro-52, anti-Mi-2b), abnormal nailfold capillaroscopy with a scleroderma pattern, interstitial lung disease and cardiac involvement. The overall clinical picture was consistent with the diagnosis of jSSc. CONCLUSIONS: LJM can be the initial sign of underlying systemic sclerosis. Nailfold capillaroscopy may help differentiate jSSc from classical LJM in pediatric patients with T1D and finger contractures or skin induration of no clear origin. This case report provides a starting point for a novel hypothesis regarding the pathogenesis of jSSc. The association between T1D and jSSc may be more than a coincidence and could suggest a relationship between glucose metabolism, fibrosis and microangiopathy. BioMed Central 2022-09-11 /pmc/articles/PMC9465903/ /pubmed/36089600 http://dx.doi.org/10.1186/s12969-022-00741-3 Text en © The Author(s) 2022 https://creativecommons.org/licenses/by/4.0/Open AccessThis article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article's Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article's Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/ (https://creativecommons.org/licenses/by/4.0/) . The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/ (https://creativecommons.org/publicdomain/zero/1.0/) ) applies to the data made available in this article, unless otherwise stated in a credit line to the data. |
spellingShingle | Case Report Mastrangelo, Greta Meneghel, Alessandra Martini, Giorgia Moretti, Carlo Zulian, Francesco Juvenile diabetes and systemic sclerosis: just a coincidence? |
title | Juvenile diabetes and systemic sclerosis: just a coincidence? |
title_full | Juvenile diabetes and systemic sclerosis: just a coincidence? |
title_fullStr | Juvenile diabetes and systemic sclerosis: just a coincidence? |
title_full_unstemmed | Juvenile diabetes and systemic sclerosis: just a coincidence? |
title_short | Juvenile diabetes and systemic sclerosis: just a coincidence? |
title_sort | juvenile diabetes and systemic sclerosis: just a coincidence? |
topic | Case Report |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9465903/ https://www.ncbi.nlm.nih.gov/pubmed/36089600 http://dx.doi.org/10.1186/s12969-022-00741-3 |
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