scholarly journals FRI0406 CARTILAGE REPAIR ACTIVITY DURING JOINT-PRESERVING TREATMENT MAY BE ACCOMPANIED BY OSTEOPHYTE FORMATION

2020 ◽  
Vol 79 (Suppl 1) ◽  
pp. 801.1-802
Author(s):  
M. Jansen ◽  
S. Mastbergen ◽  
F. Watt ◽  
E. Willemse ◽  
T. Vincent ◽  
...  
Keyword(s):  
Clinical Improvement ◽  
Cartilage Repair ◽  
Control Group ◽  
Tibial Osteotomy ◽  
Knee Oa ◽  
Natural Progression ◽  
Significant Difference ◽  
Repair Activity ◽  
One Year

Background:Knee joint distraction (KJD) is a joint-preserving treatment to postpone total knee arthroplasty (TKA) and has shown cartilage repair and clinical improvement in patients with severe knee osteoarthritis (OA), as has high tibial osteotomy (HTO). The observed cartilage repair activity could be related to an increase in transforming growth factor-β1 (TGFβ-1), which increases in the synovial fluid (SF) during KJD treatment.1However, animal and ex vivo human studies have shown that TGFβ-1 also induce formation of osteophytes, generally seen as an OA severity hallmark. Similarly, interleukin-6 (IL-6) was observed to increase in SF during KJD treatment and may also be associated with osteophytosis.As such, we hypothesized that joint-preserving regenerative treatments demonstrating cartilage repair activity lead to general tissue (re)generation, including osteophytosis.Objectives:To analyze osteophytosis after KJD and compare this to HTO and natural progression in knee OA.Methods:63 KJD patients were included in several clinical trials, one of which was a randomized controlled trial comparing patients indicated for HTO, but treated with KJD (KJDHTO; n=23) vs. patients treated with HTO (n=46). All patients received standardized radiographs before and one and two years after treatment, used to measure osteophyte size. Only patients with measurements at baseline and two-year follow-up were included. As a control group for natural progression, untreated knee OA patients from Cohort Hip & Cohort Knee (CHECK; n=1002) were studied. Only patients who received a TKA during follow-up were included, using their last two measurements before treatment to reflect natural two-year progression (n=44).A separate group of 20 patients treated with KJD in regular care underwent SF aspirations before and after treatment, and TGFβ-1 and IL-6 levels were measured by immunoassay (Mesoscale Discovery). Unstandardized radiographs were acquired before and one year after treatment, used to score osteophytes with the revised Altman score, resulting in a 0 (normal) to 12 (severe) whole-joint score. Only patients with radiographs and SF aspirations at both baseline and one-year follow-up were included.Results:After two years, both KJD (n=58) and HTO (n=38) patients showed a significant increase in osteophyte size (+6.2mm2 and +7.0mm2 resp.; both p<0.003; figure 1), with no significant differences between the treatments (p>0.38). Untreated CHECK patients who underwent TKA did not show significant two-year osteophyte changes before treatment (+2.1mm2; p=0.207; figure 1) and showed significant differences compared with KJD and HTO groups (both p<0.044). In the KJD SF aspiration group (n=17), the Altman osteophyte score was not different at one year compared to baseline (+0.2 points; p=0.653) and there was no association between baseline biomarker values and the baseline Altman osteophyte score, or between changes in these parameters (all p≥0.28). Trichotomization of patients in groups with a decrease, no change or increase in total Altman osteophyte score indicated that there was a statistically significant difference between the three groups in changes in TGFβ-1 (p=0.044; figure 2A), but not IL-6 (p=0.898; figure 2B).Conclusion:After KJD treatment, joint space widening and clinical improvement are accompanied by osteophytosis. Similar results were observed after treatment with HTO, suggesting effects occur in regenerative joint-preserving treatments in general. The increased osteophytosis may be a bystander effect of cartilage repair activity related to intra-articular factors like TGFβ-1 and questions whether osteophytosis should necessarily be considered a hallmark of OA worsening.References:[1]Watt et al, Osteoarthritis Cartilage 2020Disclosure of Interests:Mylène Jansen: None declared, Simon Mastbergen: None declared, Fiona Watt: None declared, Elske Willemse: None declared, Tonia Vincent Consultant of: Ad hoc consultancy GSK, Mundipharma, UCB, Sander Spruijt Consultant of: Consultancy to Zimmer Biomet Inc., Pieter Emans Shareholder of: Shareholder and cofounder start-up company Chondropeptix, Roel Custers: None declared, Ronald Van Heerwaarden: None declared, Floris Lafeber Shareholder of: Co-founder and shareholder of ArthroSave BV

scholarly journals Cartilage Repair Activity during Joint-Preserving Treatment May Be Accompanied by Osteophyte Formation

2021 ◽  
Vol 11 (15) ◽  
pp. 7156
Author(s):  
Mylène P. Jansen ◽  
Simon C. Mastbergen ◽  
Fiona E. Watt ◽  
Elske J. Willemse ◽  
Tonia L. Vincent ◽  
...  
Keyword(s):  
Clinical Improvement ◽  
Cartilage Repair ◽  
Bystander Effect ◽  
Cartilage Regeneration ◽  
Joint Space ◽  
Tibial Osteotomy ◽  
Osteophyte Formation ◽  
Untreated Check ◽  
Repair Activity ◽  
One Year

Knee joint distraction (KJD) treatment has shown cartilage repair and clinical improvement in patients with osteoarthritis, as has high tibial osteotomy (HTO). Following KJD, TGFβ-1 and IL-6 were increased in synovial fluid (SF), factors related to cartilage regeneration, but also to osteophyte formation. As such, osteophyte formation after both joint-preserving treatments was studied. Radiographic osteophyte size was measured before, one year, and two years after treatment. Changes were compared with natural progression in patients from the CHECK cohort before undergoing total knee arthroplasty. An additional KJD cohort underwent SF aspiration, and one-year Altman osteophyte score changes were compared to SF-marker changes during treatment. After two years, both KJD (n = 58) and HTO (n = 38) patients showed an increase in osteophyte size (+6.2 mm2 and +7.0 mm2 resp.; both p < 0.004), with no significant differences between treatments (p = 0.592). Untreated CHECK patients (n = 44) did not show significant two-year changes (+2.1 mm2; p = 0.207) and showed significant differences with KJD and HTO (both p < 0.044). In SF aspiration patients (n = 17), there were significant differences in TGFβ-1 changes (p = 0.044), but not IL-6 (p = 0.898), between patients with a decrease, no change, or increase in osteophyte Altman score. Since KJD and HTO showed joint space widening and clinical improvement accompanied by osteophyte formation, increased osteophytosis after joint-preserving treatments may be a bystander effect of cartilage repair activity related to intra-articular factors like TGFβ-1 and raises questions regarding osteophyte formation as solely characteristic of the joint degenerative process.

scholarly journals Evaluation of Accuracy of Preoperative Planning of the Femurofibular Angle in Open-Wedge High Tibial Osteotomy for Mild Medial Knee Osteoarthritis

2021 ◽  
Vol 2021 ◽  
pp. 1-8
Author(s):  
Peizhao Wang ◽  
Xiao Wang ◽  
Xiaotao Shi ◽  
Honglue Tan
Keyword(s):  
Lower Limb ◽  
Preoperative Planning ◽  
Mechanical Axis ◽  
Weight Bearing ◽  
Tibial Osteotomy ◽  
Medial Knee ◽  
Knee Oa ◽  
Significant Difference ◽  
Wedge High Tibial Osteotomy

Objective. The purpose of this study was to evaluate the usefulness of preoperative planning of the femurofibular angle (FFA) in medial open-wedge high tibial osteotomy (OWHTO) for mild medial knee osteoarthritis. Methods. Thirty-two patients (32 knees) with mild medial knee OA were retrospectively reviewed. The patients underwent preoperative planning of the FFA for OWHTO. For preoperative planning, a full-length weight-bearing X-ray photograph of the lower limb was opened within Adobe Photoshop Software, and a targeted corrective mechanical axis line of the lower limb and its intersecting point at the lateral tibial plateau surface was drawn using rectangle selection and filling tools. A frame, which encircled the tibia and fibula, was created around the predicted osteotomy plane and then rotated until the ankle center was on the targeted mechanical axis line. Subsequently, a distal femoral condyle line and a proximal fibula axis line were drawn, and the angle between the two lines was measured and defined as the femurofibular angle (FFA). During biplane OWHTO, the preoperatively determined FFA was used to complete the correction of the mechanical axis. During follow-up, the postoperative mechanical weight-bearing line (WBL) of the lower limb, the mechanical femorotibial angle (mFTA), and the FFA were measured and compared with the preoperatively determined values. Results. The mechanical WBL shifted from a preoperative value of 25.36 ± 5.02 % to a postoperative value of 56.19 ± 0.10 % from the medial border along the mediolateral width of the tibial plateau, and it was 56.57 ± 0.08 % at the final follow-up ( P < 0.01 ). The preoperatively determined value was 56.25%, and no significant difference was found compared with postoperative week-one and final follow-up values ( P > 0.05 ). The mFTA was corrected from a preoperative varus of 4.02 ± 0.63 ° to a postoperative week-one valgus of 2.37 ± 0.28 ° , and it had a valgus of 2.48 ± 0.39 ° at the final follow-up ( P < 0.01 ). No significant difference in the valgus was found compared with the postoperative week-one, final follow-up and preoperatively determined valgus of 2.34 ± 0.26 ° ( P > 0.05 ). The postoperative week-one and final follow-up FFAs were 90.34 ± 1.53 ° and 90.33 ± 1.52 ° , respectively, and no significant difference was found compared with the preoperatively determined value of 90.12 ± 1.72 ° and the intraoperative setting value of 90.25 ± 1.67 ° ( P > 0.05 ). All corrected values were within the acceptable range of preoperative planning. Conclusion. Preoperative planning of the FFA may be useful in OWHTO for patients with mild medial knee OA. Satisfactory correction of the postoperative targeted mechanical axis line of the lower limb can be obtained.

Short-term Outcomes after High Tibial Osteotomy Aimed at Neutral Alignment Combined with Arthroscopic Centralization of Medial Meniscus in Osteoarthritis Patients

2020 ◽  
Author(s):  
Hiroki Katagiri ◽  
Yusuke Nakagawa ◽  
Kazumasa Miyatake ◽  
Koji Otabe ◽  
Toshiyuki Ohara ◽  
...  
Keyword(s):  
Knee Osteoarthritis ◽  
High Tibial Osteotomy ◽  
Medial Meniscus ◽  
Control Group ◽  
Tibial Osteotomy ◽  
Short Term ◽  
Subjective Satisfaction ◽  
Significant Difference ◽  
Neutral Alignment

Abstract Purpose To improve long-term outcomes of open-wedge high tibial osteotomy (OWHTO), procedures combining OWHTO aimed at neutral alignment and arthroscopic centralization for meniscal extrusion have been introduced. The purpose of the present study was to compare short-term clinical and radiological outcomes of medial joint space width (JSW) after the OWHTO aimed at neutral alignment with and without arthroscopic centralization for an extruded medial meniscus. Methods A retrospective review of 50 primary OWHTO patients was conducted. Thirty-five patients included for analysis after exclusion criteria was applied. Twenty-one knee osteoarthritis patients, who underwent the OWHTO with arthroscopic meniscal centralization, were included in the centralization group. Fourteen patients, who underwent solely OWHTO, were included in the control group. Lysholm knee scale, International Knee Documentation Committee (IKDC) subjective score, Knee Osteoarthritis Outcome Score (KOOS), and patient subjective satisfaction scores were recorded at the final follow-up. Radiographic changes of JSW and joint line congruence angle (JLCA) were measured 2 years postoperatively. Patient demographic data were also reviewed. Results IKDC subjective scores, KOOS subgroup scores, patient subjective satisfaction scores, and Lysholm score did not show significant differences between the two groups at the final follow-up. Change of the JSW in the centralization group was significantly greater than that in the control group 2 years postoperatively (Control group: -0.1 mm, Centralization group: 0.8 mm P =0.03). Conclusion Change of JSW after OWHTO aimed at neutral alignment with arthroscopic centralization for extruded medial meniscus was greater than solely OWHTO, and there was no significant difference in the short-term clinical outcomes between the 2 procedures at the final follow-up.

scholarly journals Intervention Effects of the Health Promotion Programme “Join the Healthy Boat” on Objectively Assessed Sedentary Time in Primary School Children in Germany

2020 ◽  
Vol 17 (23) ◽  
pp. 9029
Author(s):  
Susanne Kobel ◽  
Jens Dreyhaupt ◽  
Olivia Wartha ◽  
Sarah Kettner ◽  
Belinda Hoffmann ◽  
...  
Keyword(s):  
Screen Time ◽  
Primary School Children ◽  
Control Group ◽  
Health Promotion Programme ◽  
Randomised Study ◽  
Targeted Interventions ◽  
Promotion Programme ◽  
Significant Difference ◽  
One Year

Sedentary behaviour (SB) in children is related to negative health consequences that can track into adulthood. The programme “Join the Healthy Boat” promotes reduced screen time and a less sedentary lifestyle in schoolchildren. This study investigated the effects of the programme on children’s SB. For one year, teachers delivered the programme. A total of 231 children (7.0 ± 0.6 years) participated in the cluster-randomised study; there were 154 one year later at follow-up. Children’s SB was assessed using multi-sensor accelerometery, screen time via parental questionnaire. Effects were analysed using (linear) mixed effects regression models. At baseline, children spent 211 (±89) min daily in SB, at follow-up 259 (±109) min/day with no significant difference between the intervention (IG) and control group (CG). SB was higher during weekends (p < 0.01, for CG and IG). However, at follow-up, daily screen time decreased in IG (screen time of >1 h/day: baseline: 33.3% vs. 27.4%; follow-up: 41.2% vs. 27.5%, for CG and IG, respectively). This multi-dimensional, low-threshold intervention for one year does not seem to achieve a significant reduction in children’s SB, although screen time decreased in IG. Therefore, it should be considered that screen time cannot be the key contributor to SB and should not solely be used for changing children’s SB. However, if screen time is targeted, interventions should promote the replacement of screen time with active alternatives.

scholarly journals One-year clinical evaluation of IPS Empress CAD versus polished Celtra Duo ceramic Laminate veneers (randomized controlled clinical trial)

2021 ◽  
Vol 24 (3) ◽  
pp. 13p
Author(s):  
Hayat Elbanna ◽  
Mohammed Labib Zamzam ◽  
Jylan Fouad El-Guindy ◽  
Ahmed Soliman Idris
Keyword(s):  
Survival Rate ◽  
Fracture Resistance ◽  
Clinical Performance ◽  
Control Group ◽  
Marginal Adaptation ◽  
Anterior Teeth ◽  
Significant Difference ◽  
One Year ◽  
Laminate Veneers

Objective: To evaluate fracture resistance and survival rate of IPS Empress CAD versus Polished Celtra Duo ceramic laminate veneers. Material and Methods: Thirty-six ceramic laminate veneers were fabricated for maxillary anterior teeth. The patients were divided into two groups according to the material Group 1(control group) fabricated from IPS Empress CAD laminate veneers and group 2(intervention group) fabricated from Polished Celtra Duo laminate veneers. Standardized the same preparation with butt joint design and chamfer finish line located supra gingival were performed for all the teeth. The fabrication of the veneers was performed using Cad\Cam (Ceramill motion) machine, with software (Exocad). The veneers surfaces were treated and silanated according to the manufacture instruction of each ceramic and enamel surfaces were etched where total etch adhesive protocol was obeyed using BISCO. Follow up sessions were done every two months up to one year for each patient using dental probe and operator vision to evaluate the fracture, survival rate, marginal adaptation, sensitivity and caries. according to USPHS criteria (United States Public Health Service). This was performed by an experienced, blinded investigator. Results: Fracture resistance, marginal adaptation, retention, caries and sensitivity were evaluated according to the criteria of USPHS and we found there is no significant difference as both groups scaled zero score. Conclusion: Both IPS Empress Cad and Polished Celtra Duo laminate veneers revealed successful clinical performance in terms of fracture resistance, marginal adaptation, retention, and sensitivity after one year follow up period.   Keywords Ceramic laminate veneers; IPS Empress CAD; Celtra DUO; Clinical performance.

Abstract 12604: Effect of Psychotherapy on Quality of Life and Recurrence of Events in Patients With Recurrent Vasovagal Syncope: A Randomized Study

Circulation ◽  
2020 ◽  
Vol 142 (Suppl_3) ◽  
Author(s):  
Renata de Barros e Silva ◽  
Denise Hachul ◽  
PEDRO GABRIEL M DE BARROS E SILVA ◽  
Mauricio I Scanavacca
Keyword(s):  
Quality Of Life ◽  
Vasovagal Syncope ◽  
Intervention Group ◽  
Tilt Test ◽  
Control Group ◽  
Sf 36 ◽  
Significant Difference ◽  
One Year

Introduction: Vasovagal syncope presents a benign prognosis in terms of survival but is associated with significant impairment on quality of life (QoL). Poor emotional status contributes to the recurrence of events. Hypothesis: Among patients with recurrent vasovagal syncope, weekly sessions of psychotherapy may improve QoL and reduce the number of events during one year of follow-up. Methods: A randomized controlled trial was conducted including 10 patients with recurrent vasovagal syncope and positive tilt test. Cardiac disease and ongoing psychotherapeutic interventions were the main exclusion criteria. After randomization, half of the eligible patients underwent weekly sessions of psychotherapy during 12 months, while the control group was followed according to the standard of care, without psychotherapy. QoL by SF-36 and recurrence of syncope and pre-syncope were compared between the 2 groups and also in each group pre and post randomization. Results: All patients had a positive tilt test (80% with mixed vasovagal response), 70% were female and the mean age was 47.4 ± 11.1 years. The average rate of events before inclusion was 4.1 (± 3.4) syncopes/year and 1.26 (± 0.5) episodes of presyncope/week. The groups were well balanced at baseline and all patients completed the planned follow-up of one year. In the assessment of QoL by SF-36, comparing before and one year after randomization, there was no statistically significant difference in the control group (49.9 ± 9.8 vs 46.9 ± 3.4, P = 0.20) but there was a significant improvement in the intervention group (44.9 ± 11.9 vs 68.8 ± 7.8, P < 0.01). Comparing both groups at 12 months, patients that underwent sessions of psychotherapy had better QoL (68.8 ± 7.8 vs 46.9 ± 3.4, P < 0.01). Regarding the recurrence of syncope events, there was no significant change in the control group; however, in the intervention group there was a significant reduction in the rate of presyncope episodes per month (5.6 ± 2.1 vs 1.7 ± 0.9; P = 0.02) and also a significant reduction in the rate of syncope per year (4.6 ± 3.3 vs 1.0 ± 0.7; P = 0.04). Conclusions: In a randomized clinical trial, patients with recurrent vasovagal syncope undergoing regular psychotherapeutic intervention had less recurrence of events and improved quality of life in one year.

scholarly journals Factors that Decrease Sedentary Behavior in Community-Dwelling Elderly People: A Longitudinal Study

Medicina ◽  
2020 ◽  
Vol 56 (4) ◽  
pp. 157
Author(s):  
Yutaka Owari
Keyword(s):  
Sedentary Behavior ◽  
Elderly People ◽  
Intervention Group ◽  
Participation Rate ◽  
Community Dwelling ◽  
Control Group ◽  
Behavior Rate ◽  
Significant Difference ◽  
One Year

Background and objectives: This study was to clarify how the frequency of participation in a health class affected the reduction in sedentary behavior after two years, and whether decreases in sedentary behavior in elderly people who participated in a health class persisted two years after the end of an intervention. Materials and Methods: This study was longitudinal, and the results of a previous study conducted in 2017 were added to the findings of a different year. The participants were elderly health class members at a community dwelling in Japan who participated between 2016 and 2018. Of the 86 participants that were enrolled, the data of 80 were collected in 2016. A year later, in a 2017 follow-up, the number of participants was 80, and two years later, in a 2018 follow-up, there were 71 participants. Results: There was a significant difference with regards to the reduction of the sedentary behavior rate between two different groups (a health class participation rate of more than 75% and the other with less than 75%) two years later. However, no difference in sedentary behavior rate was found between the two groups (intervention group and control group) at a two-year follow-up, despite observing differences after one year. Conclusions: Continuous participation in a health class may help reduce sedentary behavior. After two years, “Active Guide” brochures and documents may not help in reducing sedentary behavior, despite seeing improvements after one year. Persistent social participation may have a more lasting effect than one-off interventions in reducing the sedentary behavior ratio.

Levels of Prothrombin Fragment F1+2 in Patients with Hyperhomocysteinemia and a History of Venous Thromboembolism

1997 ◽  
Vol 78 (05) ◽  
pp. 1327-1331 ◽  
Author(s):  
Paul A Kyrle ◽  
Andreas Stümpflen ◽  
Mirko Hirschl ◽  
Christine Bialonczyk ◽  
Kurt Herkner ◽  
...  
Keyword(s):  
Venous Thromboembolism ◽  
Thrombin Generation ◽  
Oral Anticoagulant Therapy ◽  
Oral Anticoagulants ◽  
Control Group ◽  
Prothrombin Fragment ◽  
Hemostatic System ◽  
Significant Difference ◽  
System Activation ◽  
One Year

SummaryIncreased thrombin generation occurs in many individuals with inherited defects in the antithrombin or protein C anticoagulant pathways and is also seen in patients with thrombosis without a defined clotting abnormality. Hyperhomocysteinemia (H-HC) is an important risk factor of venous thromboembolism (VTE). We prospectively followed 48 patients with H-HC (median age 62 years, range 26-83; 18 males) and 183 patients (median age 50 years, range 18-85; 83 males) without H-HC for a period of up to one year. Prothrombin fragment Fl+2 (Fl+2) was determined in the patient’s plasma as a measure of thrombin generation during and at several time points after discontinuation of secondary thromboprophylaxis with oral anticoagulants. While on anticoagulants, patients with H-HC had significantly higher Fl+2 levels than patients without H-HC (mean 0.52 ± 0.49 nmol/1, median 0.4, range 0.2-2.8, versus 0.36 ± 0.2 nmol/1, median 0.3, range 0.1-2.1; p = 0.02). Three weeks and 3,6,9 and 12 months after discontinuation of oral anticoagulants, up to 20% of the patients with H-HC and 5 to 6% without H-HC had higher Fl+2 levels than a corresponding age- and sex-matched control group. 16% of the patients with H-HC and 4% of the patients without H-HC had either Fl+2 levels above the upper limit of normal controls at least at 2 occasions or (an) elevated Fl+2 level(s) followed by recurrent VTE. No statistical significant difference in the Fl+2 levels was seen between patients with and without H-HC. We conclude that a permanent hemostatic system activation is detectable in a proportion of patients with H-HC after discontinuation of oral anticoagulant therapy following VTE. Furthermore, secondary thromboprophylaxis with conventional doses of oral anticoagulants may not be sufficient to suppress hemostatic system activation in patients with H-HC.

RELATED FACTORS TO RELAPSE IN DEPRESSED PATIENTS AFTER COGNITIVE BEHAVIOURAL THERAPY DURING ONE YEAR PROSPECTIVE FOLLOW-UP

2011 ◽  
pp. 13-19
Author(s):  
Nhu Minh Hang Tran ◽  
Huu Cat Nguyen ◽  
Dang Doanh Nguyen ◽  
Van Luong Ngo ◽  
Vu Hoang Nguyen ◽  
...  
Keyword(s):  
Cognitive Behavioral Therapy ◽  
Relapse Rate ◽  
Behavioral Therapy ◽  
Cognitive Behavioral ◽  
Control Group ◽  
Controlled Clinical Trial ◽  
Related Factors ◽  
Depressed Patients ◽  
One Year

Objectives: To determine factors impact on the relapse in depressed patients treated with Cognitive Behavioral Therapy (CBT) during one year follow-up. Materials and Methods: 80 depressed patients divided into two groups, group 1: included 40 patients treated with CBT; group 2: 40 patients on amitriptyline. Non-randomized controlled clinical trial, opened, longiditual and prospective research. Results and Conclusions: relapse rate after CBT during 1 year follow-up is 10% (compared to 25% in control group), related factors to relapse rate in depression after CBT are age and education. Shared predictors between 2 groups are severity and recurrence of depression. Key words: Depression, relapse, Cognitive Behavioral Therapy (CBT)

scholarly journals A206 EVOLUTION OF PEDIATRIC AUTOIMMUNE CHOLANGITIS AND PRIMARY SCLEROSING CHOLANGITIS INTO ADULTHOOD

2021 ◽  
Vol 4 (Supplement_1) ◽  
pp. 234-236
Author(s):  
P Willems ◽  
J Hercun ◽  
C Vincent ◽  
F Alvarez
Keyword(s):  
Primary Sclerosing Cholangitis ◽  
Sclerosing Cholangitis ◽  
Response To Treatment ◽  
Similar Proportion ◽  
Autoimmune Cholangitis ◽  
Significant Difference ◽  
One Year ◽  
Liver Tests

Abstract Background The natural history of primary sclerosing cholangitis (PSC) in children seems to differ from PSC in adults. However, studies on this matter have been limited by short follow-up periods and inconsistent classification of patients with autoimmune cholangitis (AIC) (or overlap syndrome). Consequently, it remains unclear if long-term outcomes are affected by the clinical phenotype. Aims The aims of this is study are to describe the long-term evolution of PSC and AIC in a pediatric cohort with extension of follow-up into adulthood and to evaluate the influence of phenotype on clinical outcomes. Methods This is a retrospective study of patients with AIC or PSC followed at CHU-Sainte-Justine, a pediatric referral center in Montreal. All charts between January 1998 and December 2019 were reviewed. Patients were classified as either AIC (duct disease on cholangiography with histological features of autoimmune hepatitis) or PSC (large or small duct disease on cholangiography and/or histology). Extension of follow-up after the age of 18 was done for patients followed at the Centre hospitalier de l’Université de Montréal. Clinical features at diagnosis, response to treatment at one year and liver-related outcomes were compared. Results 40 patients (27 PSC and 13 AIC) were followed for a median time of 71 months (range 2 to 347), with 52.5% followed into adulthood. 70% (28/40) had associated inflammatory bowel disease (IBD) (78% PSC vs 54% AIC; p=0.15). A similar proportion of patients had biopsy-proven significant fibrosis at diagnosis (45% PSC vs 67% AIC; p=0.23). Baseline liver tests were similar in both groups. At diagnosis, all patients were treated with ursodeoxycholic acid. Significantly more patients with AIC (77% AIC vs 30 % PSC; p=0.005) were initially treated with immunosuppressive drugs, without a significant difference in the use of Anti-TNF agents (0% AIC vs 15% PSC; p= 0.12). At one year, 55% (15/27) of patients in the PSC group had normal liver tests versus only 15% (2/13) in the AIC group (p=0.02). During follow-up, more liver-related events (cholangitis, liver transplant and cirrhosis) were reported in the AIC group (HR=3.7 (95% CI: 1.4–10), p=0.01). Abnormal liver tests at one year were a strong predictor of liver-related events during follow-up (HR=8.9(95% CI: 1.2–67.4), p=0.03), while having IBD was not (HR=0.48 (95% CI: 0.15–1.5), p=0.22). 5 patients required liver transplantation with no difference between both groups (8% CAI vs 15% CSP; p=0.53). Conclusions Pediatric patients with AIC and PSC show, at onset, similar stage of liver disease with comparable clinical and biochemical characteristics. However, patients with AIC receive more often immunosuppressive therapy and treatment response is less frequent. AIC is associated with more liver-related events and abnormal liver tests at one year are predictor of bad outcomes. Funding Agencies None

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