Cargando…
Capillary microscopy is a potential screening method for connective tissue disease in children with Raynaud’s phenomenon
BACKGROUND: Nailfold capillary microscopy (NCM) is a cornerstone in the diagnosis of Systemic Sclerosis (SSc) in adulthood. Although Raynaud’s phenomenon (RP) is very common in childhood, studies on diagnostic methods to differentiate between primary RP (PRP) and secondary RP (SRP) at a young age ar...
Autores principales: | , , , , , , |
---|---|
Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
BioMed Central
2022
|
Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8822798/ https://www.ncbi.nlm.nih.gov/pubmed/35135560 http://dx.doi.org/10.1186/s12969-022-00671-0 |
_version_ | 1784646674903203840 |
---|---|
author | Farenhorst, Claudette A. Roon, Anniek M. Gessel, Anne I. Stel, Alja J. Bootsma, Hendrika Armbrust, Wineke Mulder, Douwe J. |
author_facet | Farenhorst, Claudette A. Roon, Anniek M. Gessel, Anne I. Stel, Alja J. Bootsma, Hendrika Armbrust, Wineke Mulder, Douwe J. |
author_sort | Farenhorst, Claudette A. |
collection | PubMed |
description | BACKGROUND: Nailfold capillary microscopy (NCM) is a cornerstone in the diagnosis of Systemic Sclerosis (SSc) in adulthood. Although Raynaud’s phenomenon (RP) is very common in childhood, studies on diagnostic methods to differentiate between primary RP (PRP) and secondary RP (SRP) at a young age are scarce. The aim of this study was to determine the value of NCM in differentiating between PRP and SRP in children and adolescents with RP. METHODS: In this nested case–control study, 83 patients diagnosed with RP and having underwent NCM in childhood were retrospectively included. Based on whether they were diagnosed with a connective tissue disease (CTD) during follow-up, patients were classified as PRP or SRP. NCM was performed by a vascular technician. PRP and SRP patients were compared on demographics, NCM and serology. Variables associated with SRP were included in a multivariate logistic regression model. Predictive values were calculated for NCM, ANA positivity and the combination of NCM and ANA positivity. RESULTS: At the time of the NCM, the mean age of the RP patients was 15.4 ± 2.3 years. Of these patients, 78.3% were classified as PRP and 21.7% as SRP at mean follow-up of 6.4 ± 3.20 years. CTDs were miscellaneous, with only one patient having developed SSc. Of the NCM parameters, only capillary loss was associated with SRP (p = 0.01). In a multivariate logistic regression model including ANA, capillary loss was not a predictor of SRP. In a model without ANAs, capillary loss was an independent predictor (OR = 3.98, CI 95% 1.22–12.99). Capillary loss had a sensitivity of 44.4% and a specificity of 84.4% for SRP. ANA combined with capillary loss had a sensitivity of 66.7% and a specificity of 85.7%. CONCLUSION: Whereas RP in adulthood is most strongly associated with SSc, children with RP seem to be at risk for developing other CTDs with less apparent NCM abnormalities. Of all NCM findings, only capillary loss was predictive for SRP. NCM did not add to the predictive value of ANA screening. However, with a specificity of 84.4% and being non-invasive, NCM shows potential as a screening method for SRP. More research with a larger study population is required before drawing conclusions. |
format | Online Article Text |
id | pubmed-8822798 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2022 |
publisher | BioMed Central |
record_format | MEDLINE/PubMed |
spelling | pubmed-88227982022-02-08 Capillary microscopy is a potential screening method for connective tissue disease in children with Raynaud’s phenomenon Farenhorst, Claudette A. Roon, Anniek M. Gessel, Anne I. Stel, Alja J. Bootsma, Hendrika Armbrust, Wineke Mulder, Douwe J. Pediatr Rheumatol Online J Research Article BACKGROUND: Nailfold capillary microscopy (NCM) is a cornerstone in the diagnosis of Systemic Sclerosis (SSc) in adulthood. Although Raynaud’s phenomenon (RP) is very common in childhood, studies on diagnostic methods to differentiate between primary RP (PRP) and secondary RP (SRP) at a young age are scarce. The aim of this study was to determine the value of NCM in differentiating between PRP and SRP in children and adolescents with RP. METHODS: In this nested case–control study, 83 patients diagnosed with RP and having underwent NCM in childhood were retrospectively included. Based on whether they were diagnosed with a connective tissue disease (CTD) during follow-up, patients were classified as PRP or SRP. NCM was performed by a vascular technician. PRP and SRP patients were compared on demographics, NCM and serology. Variables associated with SRP were included in a multivariate logistic regression model. Predictive values were calculated for NCM, ANA positivity and the combination of NCM and ANA positivity. RESULTS: At the time of the NCM, the mean age of the RP patients was 15.4 ± 2.3 years. Of these patients, 78.3% were classified as PRP and 21.7% as SRP at mean follow-up of 6.4 ± 3.20 years. CTDs were miscellaneous, with only one patient having developed SSc. Of the NCM parameters, only capillary loss was associated with SRP (p = 0.01). In a multivariate logistic regression model including ANA, capillary loss was not a predictor of SRP. In a model without ANAs, capillary loss was an independent predictor (OR = 3.98, CI 95% 1.22–12.99). Capillary loss had a sensitivity of 44.4% and a specificity of 84.4% for SRP. ANA combined with capillary loss had a sensitivity of 66.7% and a specificity of 85.7%. CONCLUSION: Whereas RP in adulthood is most strongly associated with SSc, children with RP seem to be at risk for developing other CTDs with less apparent NCM abnormalities. Of all NCM findings, only capillary loss was predictive for SRP. NCM did not add to the predictive value of ANA screening. However, with a specificity of 84.4% and being non-invasive, NCM shows potential as a screening method for SRP. More research with a larger study population is required before drawing conclusions. BioMed Central 2022-02-08 /pmc/articles/PMC8822798/ /pubmed/35135560 http://dx.doi.org/10.1186/s12969-022-00671-0 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 | Research Article Farenhorst, Claudette A. Roon, Anniek M. Gessel, Anne I. Stel, Alja J. Bootsma, Hendrika Armbrust, Wineke Mulder, Douwe J. Capillary microscopy is a potential screening method for connective tissue disease in children with Raynaud’s phenomenon |
title | Capillary microscopy is a potential screening method for connective tissue disease in children with Raynaud’s phenomenon |
title_full | Capillary microscopy is a potential screening method for connective tissue disease in children with Raynaud’s phenomenon |
title_fullStr | Capillary microscopy is a potential screening method for connective tissue disease in children with Raynaud’s phenomenon |
title_full_unstemmed | Capillary microscopy is a potential screening method for connective tissue disease in children with Raynaud’s phenomenon |
title_short | Capillary microscopy is a potential screening method for connective tissue disease in children with Raynaud’s phenomenon |
title_sort | capillary microscopy is a potential screening method for connective tissue disease in children with raynaud’s phenomenon |
topic | Research Article |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8822798/ https://www.ncbi.nlm.nih.gov/pubmed/35135560 http://dx.doi.org/10.1186/s12969-022-00671-0 |
work_keys_str_mv | AT farenhorstclaudettea capillarymicroscopyisapotentialscreeningmethodforconnectivetissuediseaseinchildrenwithraynaudsphenomenon AT roonanniekm capillarymicroscopyisapotentialscreeningmethodforconnectivetissuediseaseinchildrenwithraynaudsphenomenon AT gesselannei capillarymicroscopyisapotentialscreeningmethodforconnectivetissuediseaseinchildrenwithraynaudsphenomenon AT stelaljaj capillarymicroscopyisapotentialscreeningmethodforconnectivetissuediseaseinchildrenwithraynaudsphenomenon AT bootsmahendrika capillarymicroscopyisapotentialscreeningmethodforconnectivetissuediseaseinchildrenwithraynaudsphenomenon AT armbrustwineke capillarymicroscopyisapotentialscreeningmethodforconnectivetissuediseaseinchildrenwithraynaudsphenomenon AT mulderdouwej capillarymicroscopyisapotentialscreeningmethodforconnectivetissuediseaseinchildrenwithraynaudsphenomenon |