Juvenile diabetes and systemic sclerosis: just a coincidence?
Cheiroarthropathy
Dipeptidyl peptidase-4
Limited joint mobility
Microangiopathy
Nailfold capillaroscopy
Sclerodactyly
Journal
Pediatric rheumatology online journal
ISSN: 1546-0096
Titre abrégé: Pediatr Rheumatol Online J
Pays: England
ID NLM: 101248897
Informations de publication
Date de publication:
11 Sep 2022
11 Sep 2022
Historique:
received:
29
06
2022
accepted:
06
09
2022
entrez:
11
9
2022
pubmed:
12
9
2022
medline:
14
9
2022
Statut:
epublish
Résumé
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. 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. 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.
Sections du résumé
BACKGROUND
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
METHODS
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
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.
Identifiants
pubmed: 36089600
doi: 10.1186/s12969-022-00741-3
pii: 10.1186/s12969-022-00741-3
pmc: PMC9465903
doi:
Types de publication
Case Reports
Journal Article
Langues
eng
Sous-ensembles de citation
IM
Pagination
81Informations de copyright
© 2022. The Author(s).
Références
Joint Bone Spine. 2021 Jan;88(1):105048
pubmed: 32653653
Reumatologia. 2017;55(2):104-107
pubmed: 28539684
Diabetes Care. 2007 Aug;30(8):1964-7
pubmed: 17519431
Postgrad Med. 1999 Feb;105(2):99-101, 105-6
pubmed: 10026706
Arthritis Rheum. 2013 Nov;65(11):2737-47
pubmed: 24122180
J Rheumatol. 2012 Mar;39(3):532-8
pubmed: 22247345
Arthritis Rheumatol. 2020 Jan;72(1):137-149
pubmed: 31350829
Am Rev Respir Dis. 1984 Nov;130(5):930-2
pubmed: 6497171
Arch Dis Child. 1992 Jan;67(1):96-9
pubmed: 1739347
An Bras Dermatol. 2017 Jan-Feb;92(1):8-20
pubmed: 28225950
J Intern Med. 1993 Aug;234(2):189-93
pubmed: 8340742
Arthritis Rheum. 2007 Mar 15;57(2):203-12
pubmed: 17330294
Diabet Med. 2019 Aug;36(8):939-947
pubmed: 30920669
Rheumatol Int. 2010 Apr;30(6):793-5
pubmed: 19506880
Am J Clin Dermatol. 2005;6(4):225-37
pubmed: 16060710
Int J Clin Pract. 2012 Apr;66(4):374-7
pubmed: 22248162
Ann Rheum Dis. 2015 Nov;74(11):1968-75
pubmed: 24919467
J Intern Med. 1994 Aug;236(2):215-23
pubmed: 8046322
Pediatr Rheumatol Online J. 2019 Oct 16;17(1):68
pubmed: 31619252
J Clin Endocrinol Metab. 2013 Feb;98(2):466-73
pubmed: 23316085
Diabetes Metab. 1996 Jun;22(3):192-6
pubmed: 8697307
Rheumatology (Oxford). 2006 May;45(5):614-20
pubmed: 16368732
Diabetes Care. 2014 Jul;37(7):1863-9
pubmed: 24722493
Arch Dis Child. 2005 Oct;90(10):1039-44
pubmed: 16177159
World J Diabetes. 2015 Aug 10;6(9):1108-12
pubmed: 26265997
Front Immunol. 2019 Jun 25;10:1352
pubmed: 31293569
Arthritis Rheum. 1982 Nov;25(11):1357-61
pubmed: 6753855