Large language model may assist diagnosis of SAPHO syndrome by bone scintigraphy.

OBJECTIVE: In this study, we employed a large language model to evaluate the diagnostic efficacy of radiology reports of bone scintigraphy in the context of identifying SAPHO syndrome, and further examined the potential of such a model to augment the diagnostic procedure. METHODS: Imaging data and clinical information of 151 patients (105/46 women/men, mean age: 53.5 years) who underwent bone scintigraphy for suspected SAPHO syndrome between January 2007 and December 2022 were retrospectively reviewed. ChatGPT-4.0 was used as the large language model. The diagnostic performance of the large language model was verified by comparing the cases judged to have SAPHO syndrome that fulfilled Kahn's classification criteria based on a combination of concise radiology reports and skin lesions such as palmoplantar pustulosis, with cases diagnosed with SAPHO syndrome by rheumatologists based on all clinical information. The diagnostic performance of the large language model was verified. RESULTS: The diagnostic accuracy of a large language model for analyzing bone scintigraphy radiology reports in conjunction with information about skin symptoms, such as palmoplantar pustulosis, achieved a sensitivity of 83.5%, specificity of 69.4%, and an overall accuracy of 76.8%. DISCUSSION: While this research is an initial endeavor dedicated to the utilization of a substantial language model in the creation of a database for imaging diagnostics of rheumatic conditions, it exhibits commendable diagnostic accuracy, particularly for diseases with a wide range of symptoms like SAPHO syndrome, indicating a positive outlook for subsequent studies. CONCLUSION: This research indicates the prospective value of extensive language models in scrutinizing radiology accounts from bone scintigraphy for the diagnosis of SAPHO syndrome.

Pyoderma gangrenosum after surgery for forefoot deformity in a patient with rheumatoid arthritis: A case report.

Pyoderma gangrenosum (PG) is a rare inflammatory skin disease characterised by skin ulcers that are associated with autoimmune diseases. Although the effectiveness of immunosuppression with glucocorticoids and tumour necrosis factor inhibitors in treating PG has been reported, the utility of negative-pressure wound therapy (NPWT) for severe ulcerative lesions in patients with PG remains controversial. Herein, we report the case of a 76-year-old woman with rheumatoid arthritis who developed PG after undergoing surgery for a forefoot deformity. The patient showed improvement in deep ulcer lesions through NPWT while receiving treatment with abatacept and systemic glucocorticoids. Subsequent topical glucocorticoid therapy led to the remission of the PG. This case suggests that NPWT, when used under immunosuppressive conditions, does not exacerbate the pathergy and may be beneficial for treating severe ulcerative PG.

Assessment of clinical and radiological characteristics of Japanese patients with SAPHO syndrome.

OBJECTIVE: This study aimed to analyse the radiological characteristics and clinical diversity of Japanese patients with synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome, a heterogeneous disorder. METHODS: Radiographs and clinical information from 115 Japanese patients (female/male: 81/34, mean age at onset: 48.7 years) diagnosed with SAPHO syndrome between January 2007 and December 2020 were retrospectively reviewed. Additionally, the treatment for SAPHO syndrome was explored. RESULTS: Among the 115 patients, 70 patients had complications, including palmoplantar pustulosis, acne, or psoriasis. Imaging studies included bone scintigraphy, magnetic resonance imaging, computed tomography, and positron emission tomography in 71, 58, 70, and 23 patients, respectively. The most frequent lesions were arthritis and hyperostosis of the sternoclavicular joints in 96 patients; spinal lesions, including sacroiliac arthritis were observed in 85 patients. Peripheral aseptic osteitis was observed in 22 patients, and the tibia was involved in 12. The treatments consisted of analgesics, bisphosphonates, conventional synthetic disease-modifying anti-rheumatic drugs, and biologics (tumour necrosis factor inhibitors and interleukin-23p19 inhibitors) in 85, 15, 23, and 10 patients (8 and 2 patients), respectively. CONCLUSION: Sternoclavicular hyperostosis and pustulosis are frequently observed in patients with SAPHO syndrome. Biological agents were more frequently used in patients with peripheral osteitis and arthritis.

Two-stage revision knee arthroplasty for metallosis in a patient with rheumatoid arthritis: A case report.

We report a successful case of two-stage revision total knee arthroplasty performed for treating painless metallosis after total knee arthroplasty with a metal-backed patella. A 63-year-old woman diagnosed with rheumatoid arthritis underwent left total knee arthroplasty with a metal-backed patella at 32 years of age. The patient did not have knee pain; however, knee joint swelling, a strange noise, and pigmentation were reported 4 years ago. Radiographs showed cloud and metal-line signs anteriorly and posteriorly at the femoral condyle. Therefore, a two-stage surgery was performed for infection prevention and ease of performing posterior synovectomy. The patient underwent initial synovectomy via a posterior approach, followed by anterior synovectomy and revision total knee arthroplasty. Synovectomy was performed well without perioperative infection or failure of wound healing. In cases with metallosis after total knee arthroplasty, the two-stage revision total knee arthroplasty should be considered, depending on the degree of synovial proliferation and the risk of complications.

Five cases of seronegative persistent inflammatory monoarthritis improved with biological therapy.

Persistent inflammatory monoarthritis is defined as inflammation of one joint that continues for longer than 3 months. Most cases remain as nonspecific arthritis after several years. Persistent inflammatory monoarthritis is difficult to diagnose and treat in the early stage because there are no criteria for diagnosis and treatment. We report five seronegative persistent inflammatory monoarthritis cases that affected the left knee, right knee, left knee, left ankle, and right knee. All patients underwent joint punctures; two patients received steroid injections in the affected joint. The bacterial and mycobacterial culture were negative in all patients. Two patients received oral steroids, and two patients were administered nonsteroidal anti-inflammatory drugs; however, their symptoms did not improve, and one patient experienced progression of joint destruction. We investigated the usefulness of biological disease-modifying antirheumatic drugs for the treatment of seronegative persistent inflammatory monoarthritis. We obtained a remarkable improvement effect and prevented the advance of joint destruction.

Interstitial lung disease in a woman with rheumatoid arthritis treated with denosumab: A case report.

The present report describes the case of an 84 year old female Japanese patient with rheumatoid arthritis (RA) who experienced exacerbation of interstitial lung disease (ILD) after denosumab (Dmab) treatment. The onset of RA occurred in 2008, and the patient had been treated with intravenous or subcutaneous injection of tocilizumab since 2009. In July 2013, she experienced a lumbar vertebral fracture and began treatment with 60 mg Dmab injection every 6 months in January 2014. The patient had a history of mild ILD and was evaluated for ILD by chest computed tomography (CT) imaging prior to the start of Dmab use. The vertebral fracture did not recur after the initiation of Dmab treatment, and her osteoporosis was successfully treated. However, she expressed a concern of exacerbations of cough and respiratory discomfort that had occurred since September 2019. The chest CT image in November 2015 showed minor ILD progression, whereas the image in September 2019 showed severe exacerbation of ILD. To treat this exacerbation, 10 mg of methylprednisolone and 2.5 mg of tacrolimus were administered, and Dmab was discontinued. The patient was subsequently switched to oral bisphosphonate. The patient's respiratory discomfort and the finding of interstitial lung lesion in CT imaging improved after Dmab discontinuation. This case showed that exacerbation of ILD may occur after Dmab treatment, and physicians should consider the risks of Dmab-related ILD in patients with RA complicated by ILD.

Mathematical model for histogram analysis of dynamic contrast-enhanced MRI: A method to evaluate the drug treatment response in rheumatoid arthritis.

PURPOSE: To evaluate the effectiveness of a mathematical model for histogram analysis of DCE-MRI in distinguishing responders from non-responders during RA drug treatment. METHOD: Twenty-three consecutive RA patients with clinically active inflammation prospectively underwent DCE-MRI at baseline and after treatment. Manual segmentation of the enhanced synovium was performed on the last phase of DCE-MRI. The voxel-based contrast enhancement was calculated in each phase to obtain 75th percentile values. Kinetic curves made from the 75th percentile values were fitted to mathematical model as follows, ΔS(t) = A(1 - e-αt)e-ßt, where A is the upper limit of signal intensity (%), α (sec-1) is the rate of signal increase, and ß (sec-1) is the rate of signal decrease during washout. AUC30 was calculated by integration of 30 s. SER was calculated as the signal intensity at the initial time point (t = 60) relative to the delayed time point (t = 300). The volumes of enhanced synovium (sum of the number of voxels) were also calculated. RESULTS: After treatment, α, Aα, AUC30 and SER were significantly lower in the responder group than in the non-responder group (p = 0.033, 0.024, 0.015, and 0.007). The p value of SER was lowest. Aα, AUC30, and the volume of enhanced synovium had significantly larger changes from baseline to after treatment in the responder group than in the non-responder group (p = 0.045, 0.017, and 0.008). The volume of enhanced synovium had the lowest p value. CONCLUSIONS: SER after treatment and change in the volume of enhanced synovium might be effective for distinguishing responders from non-responders.

The Effect of Teriparatide for the Treatment of Multiple Spontaneous Clinical Vertebral Fractures after Discontinuation of Denosumab in a Female Patient with Rheumatoid Arthritis: A Case Report.

Discontinuation of denosumab is associated with the risk of rebound in bone turnover and rebound-associated spontaneous clinical vertebral fractures. This case report presents an 86-year-old woman with rheumatoid arthritis who experienced rebound-associated spontaneous clinical vertebral fractures at 9 months after denosumab discontinuation. Following 5-year bisphosphonate treatment, the patient had 9 injections of 60-mg denosumab every 6 months. Because of tooth extraction, denosumab treatment was discontinued, and raloxifene was administered. At 9 months after the last denosumab injection, the patient experienced severe low back pain. Magnetic resonance imaging (MRI) and radiograph demonstrated clinical fracture at the fourth lumbar vertebra. MRI performed at 3 months after first fracture showed two additional fractures at the second and third lumbar vertebrae. Teriparatide was administered for management of rebound-associated spontaneous clinical, multiple vertebral fractures. Teriparatide was effective for accelerating the fracture healing and suppressing the occurrence of new fractures. However, 2-year treatment of teriparatide did not have suppressive effect of rebound in bone turnover and general bone loss. This case suggested that teriparatide was effective for suppression of new rebound-associated spontaneous clinical vertebral fractures, but not effective in prevention of general bone loss after denosumab discontinuation.

Effect of interleukin-17A inhibitor in Japanese patients with psoriatic arthritis compared with tumor necrosis factor-alpha inhibitor.

OBJECTIVES: The patient of severe psoriatic arthritis (PsA) is mainly treated with oral methotrexate, ciclosporin, and anti-tumor necrosis factor-alpha inhibitors (TNFi). Recently, anti-interleukin-17A inhibitors (IL-17Ai) have been used in the treatment of PsA. This study aimed to evaluate the efficacy and safety of IL-17Ai in Japanese patients with PsA compared with those of TNFi. METHODS: This was a longitudinal and retrospective study. The study population included 31 Japanese patients with PsA. All enrolled patients fulfilled the Classification Criteria for Psoriatic Arthritis. All patients were treated with TNFi or IL-17Ai. The assessed clinical manifestations were C-reactive protein (CRP)-based Disease Activity Score in 28 Joints (DAS28-CRP), disease activity in psoriatic arthritis (DAPSA), 20% achievement of American College of Rheumatology core set, swollen joint count (SJC), tender joint count (TJC), and visual analog scale (VAS). Functional ability of patients with PsA was analyzed using the modified health assessment questionnaire (mHAQ) score. We evaluated the parameters at baseline and weeks 12, 24, and 52. RESULTS: The change in SJC, TJC, VAS, mHAQ, and DAPSA had no significant difference at weeks 12, 24, and 52. The improvements of CRP and DAS28-CRP were significantly higher in TNFi group only at week 12. The biologics retention rate was significantly higher in TNFi group by the log-rank test. No critical adverse events occurred. CONCLUSIONS: Our study presented that IL-17Ai had treatment effects comparable to TNFi. IL-17Ai might have the potential to become an alternative to the previous drug, but more large-scale studies are expected.

Effect of denosumab switched from bisphosphonates in preventing joint destruction in postmenopausal rheumatoid arthritis patients with anti-cyclic citrullinated peptide antibodies.

INTRODUCTION: This study aimed to determine the effects of denosumab treatment on the joint destruction of Japanese females with rheumatoid arthritis (RA) and anti-cyclic citrullinated peptide (CCP) antibodies. MATERIALS AND METHODS: This retrospective longitudinal study included 56 patients treated with denosumab and 50 patients treated with bisphosphonate. All participants were positive for anti-CCP antibodies. All patients also had a history of osteoporosis treatment with bisphosphonate, which was either continued or switched to 60 mg of subcutaneous denosumab injection every 6 months. To assess the progression of joint destruction, hand and foot radiographs were taken, and changes in modified total Sharp score (mTSS), erosion score (ERO), and joint space narrowing score (JSN) were evaluated at 12 months and 24 months. The changes in BMD of the lumbar spine and hip were also assessed at 12 months. RESULTS: At 12 months, there were significant differences in the change of ERO (p = 0.015) and mTSS (p = 0.01). Similarly, there were significant differences in the change of ERO (p = 0.013) and mTSS (p = 0.003) at 24 months. In contrast, no significant difference was observed in the changes of JSN and clinical parameters. There were significant differences in the changes in BMD in the femoral neck (p = 0.011) and total hip (p = 0.012). CONCLUSION: Denosumab treatment might be effective for the inhibition of bone erosion progression in the patients with RA, and it potentially contributes to the treatment of osteoporosis and prevention of destructive arthritis in patients with switching treatment from bisphosphonate.

Psoriatic arthritis with skin lesions localized to the scalp: A case report.

A 66-year-old man with a 2-year history of suspected scalp eczema with excessive dandruff developed painful swollen joints in the extremities. Four months after developing polyarthritis and polydactylitis, eczema gradually spread to the face. He was referred to our hospital for intractable scalp and facial eczema and polyarthritis. Based on the appearance of the head and facial skin lesions, psoriasis was suspected. Treatment with apremilast (a phosphodiesterase-4-inhibitor) was initiated, which swiftly alleviated the skin lesions. The joint deformities persisted, but the pain in the joints disappeared. This case implies that psoriatic arthritis should be suspected even if psoriatic skin lesions are localized to the scalp.

Tocilizumab Was Effective in Repairing the Large Geode in a Patient with Rheumatoid Arthritis.

Rheumatoid arthritis is characterized by multiple chronic arthritis subsequently inducing joint destruction. Although subchondral geode is a well-known feature of high-disease activity, a large geode is rare. Moreover, the treatment effect of biologic agents in the repair of large geode has not been reported. The present report shows the significant effect of interleukin-6 receptor blocker, tocilizumab, in repairing the large geode in the left humeral lateral epicondyle. This case implies that tocilizumab might be an effective treatment in patients with rheumatoid arthritis even with large geode.

Evaluation of risk factors of vertebral fracture in Japanese female patients with glucocorticoid-induced osteoporosis.

BACKGROUND: Glucocorticoid-induced osteoporosis and vertebral fracture are common complications in patients on glucocorticoid treatment for rheumatological diseases. The present study aimed to identify the risk factors of vertebral fracture in Japanese female patients with glucocorticoid-induced osteoporosis. METHODS: This study included 225 Japanese women with glucocorticoid-induced osteoporosis and 72 patients with postmenopausal osteoporosis. All participants were treated with bisphosphonate or denosumab for osteoporosis with active form of vitamin D for at least 3 years. The differences of clinical parameters, including age, disease duration, body mass index (BMI), bone mineral density (BMD), and the dose and treatment duration of glucocorticoid were assessed between patients with and without vertebral fracture. Multivariate logistic regression analysis was also performed to evaluate the association of vertebral fracture with clinical parameters. RESULTS: The significant differences related to age, BMD of the hip, disease duration, glucocorticoid treatment duration between patients with and without vertebral fractures were demonstrated. The present study indicated that disease duration, BMI, and the total hip BMD were independent risk factors for vertebral fractures in patients with glucocorticoid-induced osteoporosis. CONCLUSIONS: Prolonged disease duration, low BMI, and low total hip BMD could be risk factors of vertebral fracture in patients on glucocorticoid treatment for rheumatological diseases.

The effect of anti-IL-6 receptor antibody for the treatment of McH-lpr/lpr-RA1 mice that spontaneously developed destructive arthritis and enthesitis.

BACKGROUND: McH-lpr/lpr-RA1 mice are a new strain of mice which spontaneously develop destructive arthritis and enthesitis in the ankle. There is no published data that drug treatment has been trialed on these mice. This study examined the effect of the mouse anti-IL-6 receptor antibody, MR16-1, for the treatment of arthritis and enthesitis in McH-lpr/lpr-RA1 mice. METHODS: Male McH-lpr/lpr-RA1 mice were randomly divided into control and treatment groups. MR16-1 was administered from 10 weeks of age for the treatment group. Saline was applied for the control group. The drug was administered once a week, at an initial dose of 2 mg, then maintained at 0.5 mg once per week thereafter. The effects were evaluated by the histopathological synovitis score, in vivo imaging using indocyanine green liposomes, and analysis of the gene expression of inflammatory cytokines. RESULTS: Tissue analyses were carried out at 14, 17 and 20 weeks of age. The synovitis scores of treated groups were significantly lower compared with those of the control group at 14 and 17 weeks of age. The kappa coefficient was 0.77. However, progression of entheseal ossification persisted in the MR16-1 treated group. In vivo imaging using indocyanine green liposomes showed significant decreases in signal intensities of treated groups at week 14, but no significant differences were observed at week 18. Blood serum amyloid A levels in treated groups were significantly lower at 17 weeks of age. The gene expression levels of Tnf and Il17 were also significantly lower in MR16-1 treated groups. CONCLUSIONS: Administration of the anti-IL-6 receptor antibody is effective for the treatment of synovitis and bone destruction of McH-lpr/lpr-RA1 mice. McH-lpr/lpr-RA1 mice may be a suitable experimental model for the development of new treatments for destructive arthritis and enthesitis. IL-6 signal blockade could contribute to the treatment of destructive arthritis, and further studies should be carried out to confirm its potential in the prevention of enthesopathy developed to ossification.

Assessment of the risk of low bone mineral density in premenopausal Japanese female patients with systemic lupus erythematosus.

BACKGROUND: The aim of this study was to assess the relationships between clinical parameters and bone mineral density (BMD) in Japanese female patients with systemic lupus erythematosus (SLE). METHODS: A total of female 136 SLE patients without menopause were retrospectively assessed to identify associations between age, disease duration, body mass index (BMI), glucocorticoid usage and disease activity and BMD based on the treatment with or without bisphosphonate. There were 71 patients treated with bisphosphonate (bisphosphonate group) and 65 patients without (non-bisphosphonate group). We evaluated the impact of age, disease duration, BMI, serologic SLE markers, glucocorticoid use on BMD of the anterior-posterior (AP) and lateral lumbar spine, total hip and femoral neck using univariate and multivariate linear regression analyses of both bisphosphonate and non-bisphosphonate groups. RESULTS: Multivariate linear regression analyses showed that in non-bisphosphonate group disease duration was negatively associated with BMD of AP spine and femoral neck, whereas in bisphosphonate group these negative associations were not present. However, multivariate linear regression analyses showed a significant relationship between BMI and BMD of the AP spine, femoral neck and total hip, regardless of bisphosphonate treatment. CONCLUSIONS: Bisphosphonate treatment eliminated the negative relationships between disease duration and the BMD of the spine and hip. AP spine and hip BMD in patients with SLE depend on BMI, regardless of bisphosphonate use. SLE serologic markers and glucocorticoid use were not negatively associated with generalized bone loss. SLE patients with low BMI have a high risk of generalized bone loss, and should be assessed and treated to prevent osteoporosis even before menopause.