TY - JOUR
T1 - Reliable detection of subtypes of nailfold capillary haemorrhages in childhood-onset systemic lupus erythematosus
AU - Bergkamp, S. C.
AU - Schonenberg-Meinema, D.
AU - Rashid, A. Nassar-Sheikh
AU - Melsens, K.
AU - Vanhaecke, A.
AU - Boumans, M. J. H.
AU - Muller, P. C. E. Hissink
AU - Cutolo, M.
AU - Kuijpers, T. W.
AU - van den Berg, J. M.
AU - Smith, V.
N1 - Funding Information: laboratory assistant in AUMC who alerted us in 2011 on the observation of "pericapillary extravasations" by microscope in SLE-patients Funding Information: Data availability statement: the data underlying this article will be shared upon reasonable request to the corresponding author. Funding: this work was supported by the ‘Stichting Steun Emma’, a Dutch foundation for the clinical researchers of the Emma children’s hospital. Vanessa Smith is a Senior Clinical Investigator of the Research Foundation - Flanders (Belgium) (FWO) [1.8.029.20N]. The FWO was not involved in study design, collection, analysis and interpretation of data, writing of the report, nor in the decision to submit the manuscript for publication. Vanessa Smith is supported by an unrestricted educational chair on systemic sclerosis of Janssen-Cilag NV. Janssen-Cilag NV was not involved in study design, collection, analysis and interpretation of data, DOI checked writing of the report, nor in the decision to submit the manuscript for publication. Competing interests: V. Smith has received speaker fees from Boehringer-Ingelheim Pharma GmbH & Co., Janssen-Cilag NV, UCB Biopharma SPRL; consultancy fees from Boehringer-Ingelheim Pharma GmbH & Co., and grant/research support from Research Foundation - Flanders (FWO), Belgian Fund for Scientific Research in Rheumatic Diseases (FWRO), Boehringer-Ingelheim Pharma GmbH & Co. and Janssen-Cilag NV. The other co-authors have declared no competing interests. Publisher Copyright: © Clinical and Experimental Rheumatology 2021.
PY - 2021/9/1
Y1 - 2021/9/1
N2 - Objective In systemic lupus erythematosus (SLE), it is necessary to obtain biomarkers that predict cardiovascular complications due to premature atherosclerosis, which is related to endothelial dysfunction. Nailfold capillary abnormalities might be a biomarker for endothelial dysfunction. In adults and children with SLE, nailfold capillary haemorrhages have shown to be significantly correlated with disease activity. Recently, different subtypes of capillary haemorrhages have been described in childhood-onset SLE (cSLE). The aim of the current study was to assess the inter-and intra-rater reliability of observations of different subtypes of haemorrhages in cSLE patients. Methods Five raters blindly evaluated 140 capillaroscopy images from 35 cSLE-patients (diagnosed according to the 2012 SLICC criteria). The images were assessed qualitatively (present or absent) and quantitatively (total number) on four different subtypes of haemorrhages: 1) punctate extravasations, 2) perivascular haemorrhage, 3) large confluent haemorrhage and 4) non-definable. As subgroups 1) and 2) were interpreted as a continuous spectrum, a post-hoc analysis with "merged"(mean) kappa/ICC was additionally calculated as one sub-group. Results Qualitative assessment showed a kappa 0.65 (95% CI: 0.60 0.70) for "punctate extravasations and perivascular haemorrhages merged"and a kappa 0.78 (95% CI: 0.72 0.83) for large confluent haemorrhages. For the quantitative assessment, ICC was 0.82 (95% CI: 0.76 0.87) for the "merged groups"and ICC 0.93 (95% CI: 0.91 0.95) for large confluent haemorrhages. Conclusion Our study shows that different subtypes of capillary haemorrhages in cSLE-patients could be reliably reproduced by different raters. This confirms our recent observation of perivascular extravasations as a subgroup of capillary haemorrhage in cSLE that might reflect endothelial dysregulation.
AB - Objective In systemic lupus erythematosus (SLE), it is necessary to obtain biomarkers that predict cardiovascular complications due to premature atherosclerosis, which is related to endothelial dysfunction. Nailfold capillary abnormalities might be a biomarker for endothelial dysfunction. In adults and children with SLE, nailfold capillary haemorrhages have shown to be significantly correlated with disease activity. Recently, different subtypes of capillary haemorrhages have been described in childhood-onset SLE (cSLE). The aim of the current study was to assess the inter-and intra-rater reliability of observations of different subtypes of haemorrhages in cSLE patients. Methods Five raters blindly evaluated 140 capillaroscopy images from 35 cSLE-patients (diagnosed according to the 2012 SLICC criteria). The images were assessed qualitatively (present or absent) and quantitatively (total number) on four different subtypes of haemorrhages: 1) punctate extravasations, 2) perivascular haemorrhage, 3) large confluent haemorrhage and 4) non-definable. As subgroups 1) and 2) were interpreted as a continuous spectrum, a post-hoc analysis with "merged"(mean) kappa/ICC was additionally calculated as one sub-group. Results Qualitative assessment showed a kappa 0.65 (95% CI: 0.60 0.70) for "punctate extravasations and perivascular haemorrhages merged"and a kappa 0.78 (95% CI: 0.72 0.83) for large confluent haemorrhages. For the quantitative assessment, ICC was 0.82 (95% CI: 0.76 0.87) for the "merged groups"and ICC 0.93 (95% CI: 0.91 0.95) for large confluent haemorrhages. Conclusion Our study shows that different subtypes of capillary haemorrhages in cSLE-patients could be reliably reproduced by different raters. This confirms our recent observation of perivascular extravasations as a subgroup of capillary haemorrhage in cSLE that might reflect endothelial dysregulation.
KW - Capillaroscopy
KW - Capillary haemorrhage
KW - Childhood-onset
KW - Systemic lupus erythematosus
UR - http://www.scopus.com/inward/record.url?scp=85115332038&partnerID=8YFLogxK
M3 - Article
C2 - 34128796
SN - 0392-856X
VL - 39
SP - 1126
EP - 1131
JO - Clinical and experimental rheumatology
JF - Clinical and experimental rheumatology
IS - 5
ER -