Anti-citrullinated Protein Antibody-positive Rheumatoid Arthritis Associated with RS3PE Syndrome-like Symptoms and an Elevated Serum Vascular Endothelial Growth Factor Level in a Patient with Myasthenia Gravis

Horai, Yoshiro; M, Honda; Nishino, Ayako; Nakashima, Yoshikazu; Suzuki, Takahisa; Kawashiri, Shin‐ya; Ichinose, Kunihiro; Tamai, Mami; Nakamura, Hideki; Motomura, Masakatsu; Origuchi, Tomoki; Kawakami, Atsushi

doi:10.2169/internalmedicine.53.1897

Cited by 8 publications

(8 citation statements)

References 10 publications

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“…Arima et al measured serum VEGF levels of three RS3PE patients and compared it with those of patients with several classic connective tissue diseases in 2005 and found several-fold increase of VEGF levels in patients with RS3PE over the controls [57]. In the same report, tumor necrosis factor α and interleukin-1 levels were not different Additional Japanese reports showed elevated VEGF levels in one patient with sarcoidosis and RS3PE, with subsequent reduction after glucocorticoid treatment [45], one patient with angioimmunoblastic T cell lymphoma [28], and another patient with RS3PE and myasthenia gravis [58]. In addition, interleukin-6 and matrix metalloproteinase-3 have been reportedly elevated in RS3PE patients with neoplasia versus those without as well [59,60].…”

Section: Advances Of Pathogenic Study In Rs3pementioning

confidence: 86%

RS3PE: Clinical and Research Development

Altman

Yao

2015

Curr Rheumatol Rep

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Remitting seronegative symmetrical synovitis with pitting edema or RS3PE is a rare elderly-onset rheumatic syndrome. Although there are overlapping clinical manifestations between RS3PE, elderly-onset rheumatoid arthritis, and polymyalgia rheumatica, RS3PE has distinct characteristics. RS3PE can be associated with neoplasia and various rheumatic conditions, suggesting that it may be heterogeneous, and is considered as a paraneoplastic rheumatic disease. The pathogenesis of RS3PE may involve vascular endothelial growth factor and infection in RS3PE based upon limited data. Patients with RS3PE without concomitant malignancy respond well to small doses of glucocorticoids and carry good prognosis.

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Section: Advances Of Pathogenic Study In Rs3pementioning

confidence: 86%

RS3PE: Clinical and Research Development

Altman

Yao

2015

Curr Rheumatol Rep

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“…Initially, some clinicians believed RS3PE to be a subset of RA, while others thought of it as a variant of PMR 1. However, Horai et al concluded that RS3PE is a distinct entity rather than a subset of other rheumatological conditions as it always presents with pitting edema, negative RF, the absence of bony erosion, an absence of the human leukocyte antigen-DRB1 genotype (commonly seen in RA) and has an excellent therapeutic response to small doses of glucocorticoids 25. RS3PE has not been proven to be a variant of PMR but does share some similarities such as presentation in an elderly population, abrupt onset, symmetrical manifestations and a good responses to low-dose glucocorticoid therapy 29.…”

Section: Discussionmentioning

confidence: 99%

“…However, we observed associations of RSPE with non-malignant and nonrheumatological condition disease entities such as diabetes mellitus type 2, pleural and pericardial effusions and autoimmune conditions like systemic lupus erythematosus and myasthenia gravis. [21][22][23][24][25] Infectious disease processes have…”

Section: Rs3pe Presenting With Other Conditionsmentioning

confidence: 99%

Remitting Seronegative Symmetrical Synovitis with Pitting Edema: A Review

Lakhmalla

Dahiya

Kichloo

et al. 2021

Journal of Investigative Medicine

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Remitting seronegative symmetrical synovitis with pitting edema (RS3PE) is a rare benign rheumatological condition characterized by sudden-onset symmetrical distal extremity edema. It can present as an isolated disease process or could be associated with other conditions. Rheumatoid factor and anticitrullinated protein antibodies are negative by definition. In current literature, there is paucity of data about the disease process. We performed a literature search using the PubMed database to identify 38 articles that met our inclusion and exclusion criteria. Our literature review focuses on the clinical picture and its diverse associations. Role of various acute phase reactants has also been outlined. There is a generalized consensus among clinicians on using moderate dose steroids for treatment. Other management options for refractory cases have also discussed. Value of several imaging modalities in diagnostic evaluation of this disease entity is touched on. Since RS3PE can be associated with other diseases, specifically cancer, timely diagnosis of this condition is necessary.

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“…RTX (1000 mg, then 500 mg every 6 months) led to the remission of both MG and RA Angelucci et al, 2010 [ 19 ] 68 F Seropositivity not specified, PR Seropositivity not specified Remission GC, AZA, ADA No impact on MG was reported. Exacerbation of MG after antirheumatic drugs withdrawal for pneumonia Comorbidity: Crohn’s disease, Uveitis Horai et al, 2014 [ 22 ] 73 M Manifestation not specified, seropositivity not specified, MM-2 ACPA + new-onset MTX, GC No impact on MG was reported Kerkeni et al, 2008 [ 23 ] 50 F Generalized, anti-AchR + , new-onset Seronegative active GC, MTX, AZA, IVIG, PEEX, RTX New-onset of MG and active RA, therapy with AZA, IVIG, and PEEX ineffective; RTX (375 mg/m 2 weekly over 4 weeks) led to remission of both MG and RA Pelachas et al, 2020 [ 24 ] 42 F Ocular, seronegative, new-onset RF + , remission MTX, ADA MG onset after 18 months of ADA and 24 months of MTX Fee et al, 2009 [ 25 ] 66 M Generalized, anti-AchR + , new-onset RF—no additional information ETN MG onset after 6 yrs of ETN, resolution after suspension Wakata et al, 2001 [ 26 ] 56 F Generalized, thymectomy, anti-AchR + , MM-0 RF + , new-onset Diphenyl sulfone auranofin loxo...…”

Section: Methodsmentioning

confidence: 99%

“…We retrieved 9 case reports of patients with concomitant RA and MG [19][20][21][22][23][24][25][26][27] and described three more cases. The clinical and therapeutic characteristics are summarized in Table 2.…”

Section: Literature Reviewmentioning

confidence: 99%

Rheumatoid arthritis and myasthenia gravis: a case-based review of the therapeutic options

et al. 2022

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Introduction Myasthenia gravis is an autoimmune disease affecting the neuromuscular junction, often associated with other autoimmune diseases, including rheumatoid arthritis. Patients with rheumatoid arthritis present an increased prevalence of myasthenia gravis compared to the general population. While these two diseases share some therapeutic options, such as glucocorticoids, methotrexate, and rituximab, there are no guidelines for treating concomitant disease. We aim to review the available evidence and to discuss the efficacy and safety of the therapeutic options in patients with rheumatoid arthritis associated with myasthenia gravis. Method We described three patients with rheumatoid arthritis associated with myasthenia gravis and we performed a systematic review of the associated literature. Results A 48-year-old man and two women (48 and 55 years old) with concomitant diagnoses of active rheumatoid arthritis and well-controlled myasthenia gravis are described. They were treated with methotrexate, leflunomide, upadacitinib, and adalimumab. None of them experienced changes in their myasthenic symptoms. We found 9 additional cases from our literature review. Methotrexate, rituximab, upadacitinib, diphenyl sulfone, auranofin, and loxoprofen sodium did not show an impact on the seven patients with previously well-controlled myasthenia. Glucocorticoids, methotrexate, and rituximab proved effective in active myasthenia gravis and arthritis. Conflicting data emerged for Tumor-necrosis factor inhibitors. Conclusions Although the available evidence remains scarce, we consider glucocorticoids, methotrexate, and rituximab as safe and effective options. The role of tumor-necrosis factor inhibitors remains uncertain. Eventually, Janus Kinase inhibitors are a novel interesting option for these patients. Key Points• To date, the only evidence on the treatment of patients with rheumatoid arthritis and concomitant myasthenia gravis derives from case reports.• Based on the review of the available case reports and on the cases we described, we consider glucocorticoids, methotrexate, and rituximab as safe and effective options, while the role of Tumor-necrosis factor inhibitors remains uncertain.• Based on the cases we described, Janus Kinase inhibitors are a novel interesting option for patients with concomitant rheumatoid arthritis and myasthenia gravis.

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Anti-citrullinated Protein Antibody-positive Rheumatoid Arthritis Associated with RS3PE Syndrome-like Symptoms and an Elevated Serum Vascular Endothelial Growth Factor Level in a Patient with Myasthenia Gravis

Cited by 8 publications

References 10 publications

RS3PE: Clinical and Research Development

RS3PE: Clinical and Research Development

Remitting Seronegative Symmetrical Synovitis with Pitting Edema: A Review

Rheumatoid arthritis and myasthenia gravis: a case-based review of the therapeutic options

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