The Spectrum of Still’s Disease: A Comparative Analysis of Phenotypic Forms in a Cohort of 238 Patients
Abstract
:1. Introduction
2. Materials and Methods
2.1. Identification and Selection of sJIA and AOSD Cases
2.2. Data Collection
2.3. Statistical Analysis
2.4. Ethics
3. Results
3.1. Characteristics of the Study Population
3.2. Comparison between the Systemic and the Chronic Patterns
3.3. Comparison between sJIA and AOSD Patients
3.4. Outcomes and Prognostic Factors
4. Discussion
5. Conclusions
Supplementary Materials
Author Contributions
Funding
Institutional Review Board Statement
Informed Consent Statement
Data Availability Statement
Acknowledgments
Conflicts of Interest
References
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Characteristics | Chronic (n = 55) | Systemic (n = 159) | Total (n = 238) | p-Values (Univariate) * | |
---|---|---|---|---|---|
Epidemiology | |||||
Men/Women | 19/36 | 74/85 | 102/136 | 0.165 | |
Median age at diagnosis, y [IQR] | 26.4 (11.0–48.1) | 26.4 (11.6–39.3) | 26.6 (10.5–43.4) | 0.827 | |
Median diagnostic delay, d [IQR] | 47 (30–200) | 30 (18–69) | 33 (19–90) | 0.018 | |
Caucasian origin, N (%) | 29/41 (70.7%) | 87/128 (68%) | 132/186 (71.0%) | 0.890 | |
sJIA, N (%) | 22 (40.0%) | 56 (35.2%) | 86/238 (36.1%) | 0.637 | |
AOSD, N (%) | 33 (60.0%) | 103 (64.8%) | 152/238 (63.9%) | 0.637 | |
Classification | |||||
criteria | ILAR, N (%) | 7/15 (46.7%) | 28/51 (54.9%) | 35/69 (50.7%) | 0.7189 |
PRINTO, N (%) | 7/13 (53.8%) | 33/51 (64.7%) | 40/72 (55.6%) | 0.530 | |
Yamaguchi, N (%) | 16/28 (57.1%) | 65/99 (65.7%) | 87/143 (60.8%) | 0.545 | |
Fautrel, N (%) | 15/28 (53.6%) | 73/99 (73.7%) | 94/143 (65.7%) | 0.070 | |
Clinical features | |||||
Fever, N (%) | 50/55 (90.9%) | 150/156 (96.2%) | 222/235 (94.5%) | 0.159 | |
Fever > 39 °C, N (%) | 29/37 (78.4%) | 122/136 (89.7%) | 168/193 (87.0%) | 0.092 | |
Joint involvement N (%) | 51/55 (92.7%) | 138/157 (87.9%) | 210/236 (89.0%) | 0.460 | |
Skin rash, N (%) | 29/53 (54.7%) | 123/156 (78.8%) | 167/233 (71.7%) | 0.001 | |
Sore throat, N (%) | 23/52 (44.2%) | 84/150 (56.0%) | 117/225 (52.0%) | 0.192 | |
Myalgia, N (%) | 12/49 (24.5%) | 54/152 (35.5%) | 72/225 (32.0%) | 0.209 | |
Lymphadenopathy, N (%) | 11/35 (31.4%) | 59/125 (47.2%) | 81/179 (45.3%) | 0.142 | |
Splenomegaly, N (%) | 1/43 (2.3%) | 24/129 (18.6%) | 29/192 (15.1%) | 0.018 | |
Hepatomegaly, N (%) | 3/43 (7.0%) | 19/126 (15.1%) | 27/188 (14.4%) | 0.271 | |
Pericarditis, N (%) | 8/52 (15.4%) | 30/158 (19.0%) | 41/233 (17.6%) | 0.706 | |
Pleurisy, N (%) | 5/55 (9.1%) | 26/159 (16.4%) | 32/238 (13.4%) | 0.273 | |
Digestive involvement, N (%) | 4/52 (7.7%) | 34/154 (22.1%) | 42/229 (18.3%) | 0.035 | |
Weight loss, N (%) | 11/32 (34.4%) | 30/107 (28.0%) | 52/157 (33.1%) | 0.639 |
Biological Characteristics | Chronic (n = 55) | Systemic (n = 159) | Total (n = 238) | p-Values (Univariate) * |
---|---|---|---|---|
WBCs, median [IQR], G/L | 14.0 (8.8–17.3) | 16.0 (12.8–20.4) | 15.9 (11.8–20.3) | 0.020 |
PMNs, median [IQR], G/L | 10.0 (6.0–13.2) | 13.0 (9.9–17.0) | 12.5 (8.6–16.3) | <0.001 |
Hemoglobin, mean (SD), g/L | 117 (±18.3) | 113 (±17.5) | 113 (±17.3) | 0.291 |
Platelets, median [IQR], G/L | 376 (300–526) | 374 (265–492) | 375 (267–492) | 0.386 |
Serum ferritin, median [IQR], µg/L | 1976 (242–5000) | 2450 (598–8616) | 2200 (541–8000) | 0.089 |
Ferritin glycosylated fraction, median [IQR], % | 19 (14–28) | 14.0 (9–22) | 15 (10–23) | 0.041 |
Ferritin glycosylated fraction <20%, N (%) | 13/26 (50.0%) | 60/84 (71.4%) | 83/125 (66.4%) | 0.043 |
CRP, median [IQR], mg/L | 131 (74–210) | 177 (104–252) | 164 (97–245) | 0.029 |
AST, median [IQR], U/L | 31 (22–48) | 42(27–86) | 39 (26–80) | 0.025 |
ALT, median [IQR], U/L | 24 (15–40) | 33 (14–87) | 30 (14–76) | 0.227 |
GGT, median [IQR], U/L | 40 (21–94) | 57 (23–161) | 55 (22–152) | 0.163 |
LDH, median [IQR], U/L | 298 (216–397) | 392 (272–570) | 381 (266–572) | 0.020 |
PT, median [IQR], % | 81 (75–93) | 76 (64–88) | 78 (68–88) | 0.075 |
Fibrinogen, mean (SD), g/L | 6.1 (±2.4) | 6.4 (±2.2) | 6.2 (±2.2) | 0.519 |
Triglycerides, median [IQR], mmol/L | 1.54 (1.11–1.88) | 1.55 (1.16–2.21) | 1.55 (1.17–2.02) | 0.614 |
Rheumatoid factor, N (%) | 3/33 (9.1%) | 7/93 (7.5%) | 14/137 (10.2%) | 0.721 |
ANA, N (%) | 15/44 (34.1%) | 24/129 (18.6%) | 42/191 (22.0%) | 0.056 |
Characteristics | sJIA (n = 86) | AOSD (n = 152) | p-Values (Univariate) * | |
---|---|---|---|---|
Epidemiology | ||||
Men/Women | 38/48 | 64/88 | 0.861 | |
Median age at diagnosis, y [IQR] | 6.6 (2.8–12.3) | 38.3 (27.1–51.7) | ||
Caucasian origin, N (%) | 46/66 (69.7%) | 86/120 (71.7%) | 0.909 | |
Evolution | ||||
Chronic, N (%) | 22 (25.6%) | 33 (21.7%) | 0.637 | |
Systemic, N (%) | 56 (65.1%) | 103 (67.8%) | 0.637 | |
Unclassified course, N (%) | 8 (9.3%) | 16 (10.5%) | 0.637 | |
Clinical features | ||||
Fever, N (%) | 81/86 (94.2%) | 141/149 (94.6%) | 1.000 | |
Joint involvement N (%) | 75/86 (87.2%) | 135/150 (90.0%) | 0.658 | |
Skin rash, N (%) | 60/84 (71.4%) | 107/149 (71.8%) | 1.000 | |
Sore throat, N (%) | 20/82 (24.4%) | 97/143 (67.8%) | <0.001 | |
Myalgia, N (%) | 18/80 (22.5%) | 54/145 (37.2%) | 0.034 | |
Lymphadenopathy, N (%) | 29/61 (47.5%) | 52/118 (44.1%) | 0.776 | |
Splenomegaly, N (%) | 12/69 (17.4%) | 17/123 (13.8%) | 0.651 | |
Hepatomegaly, N (%) | 13/69 (18.8%) | 14/119 (11.8%) | 0.264 | |
Pericarditis, N (%) | 17/84 (20.2%) | 24/149 (16.1%) | 0.538 | |
Pleurisy, N (%) | 11/86 (6.98%) | 21/152 (13.8%) | 0.980 | |
Digestive involvement, N (%) | 17/84 (20.2%) | 25/145 (17.2%) | 0.698 | |
Weight loss, N (%) | 13/48 (27.1%) | 39/109 (35.8%) | 0.377 | |
Biological | ||||
characteristics | WBCs, median [IQR], G/L | 17.4 (14.4–22.4) | 15.0 (10.9–18.9) | 0.001 |
PMNs, median [IQR], G/L | 13.4 (9.6–17.9) | 12.0 (8.5–15.3) | 0.094 | |
Serum ferritin, median [IQR], µg/L | 1076 (300–3077) | 2821 (990–10,224) | <0.001 | |
CRP, median [IQR], mg/L | 120 (82–194) | 186 (105–268) | <0.001 | |
AST, median [IQR], U/L | 33 (24–45) | 47 (27–96) | 0.004 | |
ALT, median [IQR], U/L | 14 (9–27) | 50 (21–106) | <0.001 | |
LDH, median [IQR], U/L | 391 (298–505) | 378 (242–588) | 0.767 | |
PT, median [IQR], % | 71 (58–84) | 79 (69–90) | 0.011 | |
Fibrinogen, mean (SD), g/L | 5.7 (±1.9) | 6.6 (±2.3) | 0.009 | |
Rheumatoid factor, N (%) | 2/32 (6.25%) | 12/105 (11.4%) | 0.520 | |
ANA, N (%) | 13/59 (22.4%) | 29/133 (21.8%) | 1.000 |
Present Study | Pouchot 1991 [2] | Pay 2006 [31] | Gerfaut-Valentin 2014 [28] | Sfriso 2016 [4] | Hu 2019 [29] | |
---|---|---|---|---|---|---|
Country | France | Canada | Turkey | France | Italy | China |
Case number | 238 | 62 | 120 | 57 | 245 | 517 |
Diagnosis criteria | CO | Composite | Y (AOSD) D (SJIA) | Y or F | Y | Y |
Age at onset, y | 26.6 * | 24 * | NA | 36* | 38.8 § | 37.7 § |
Women | 57% | 45% | 53% | 53% | 47% | 72% |
Fever | 95% | 100% | 94% | 95% | 93% | 100% |
Arthralgia | 89% | 100% | 95% | 95% | 93% | 73% |
Sore throat | 52% | 92% | 58% | 53% | 62% | 61% |
Skin rash | 72% | 87% | 78% | 77% | 68% | 80% |
Lymphadenopathy | 45% | 74% | 40% | 60% | 60% | 51% |
Splenomegaly | 15% | 55% | 42% | 30% | NA | 34% |
Hepatomegaly | 14% | 41% | 45% | 21% | 42% | 7% |
Pericarditis | 18% | 27% | 10% | 19% | 17% | 14% |
WBCs ≥10,000/mm3 | 81% | 94% | NA | 72% | 81% | 86% |
PMNs ≥ 80% | 58% | 88% | 71% | 77% | 70% | 78% |
Elevated liver enzymes | 54% | 76% | 55% | 66% | 54% | 62% |
SF level | ||||||
>1 × normal | 95% | NA | 89% | 82% | 80% | 96% |
>5 × normal | 73% | NA | 60% | NA | NA | 79% |
GF < 20% | 66% | NA | NA | 76% | NA | NA |
Positive ANA | 22% | 11% | NA | 8% | 10% | 9% |
Positive RF | 10% | 6% | NA | 0% | 4% | 6% |
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Neau, P.-A.; El-Jammal, T.; Javaux, C.; Fournier, N.; Chol, O.; Adelaïde, L.; Ly, K.H.; Gerfaud-Valentin, M.; Perard, L.; Fouillet-Desjonqueres, M.; Le Scanff, J.; Vignot, E.; Hot, A.; Belot, A.; Durieu, I.; Sève, P.; Jamilloux, Y. The Spectrum of Still’s Disease: A Comparative Analysis of Phenotypic Forms in a Cohort of 238 Patients. J. Clin. Med. 2022, 11, 6703. https://doi.org/10.3390/jcm11226703
Neau P-A, El-Jammal T, Javaux C, Fournier N, Chol O, Adelaïde L, Ly KH, Gerfaud-Valentin M, Perard L, Fouillet-Desjonqueres M, Le Scanff J, Vignot E, Hot A, Belot A, Durieu I, Sève P, Jamilloux Y. The Spectrum of Still’s Disease: A Comparative Analysis of Phenotypic Forms in a Cohort of 238 Patients. Journal of Clinical Medicine. 2022; 11(22):6703. https://doi.org/10.3390/jcm11226703
Chicago/Turabian StyleNeau, Pierre-Antoine, Thomas El-Jammal, Clément Javaux, Nicolas Fournier, Orlane Chol, Léopold Adelaïde, Kim Heang Ly, Mathieu Gerfaud-Valentin, Laurent Perard, Marine Fouillet-Desjonqueres, Julie Le Scanff, Emmanuelle Vignot, Arnaud Hot, Alexandre Belot, Isabelle Durieu, Pascal Sève, and Yvan Jamilloux. 2022. "The Spectrum of Still’s Disease: A Comparative Analysis of Phenotypic Forms in a Cohort of 238 Patients" Journal of Clinical Medicine 11, no. 22: 6703. https://doi.org/10.3390/jcm11226703