The following is a summary of “Is Satoyoshi syndrome an autoimmune disease? A systematic review,” published in the July 2023 issue of Rheumatology by Montanaro, et al.
For a systematic review, researchers sought to evaluate the evidence supporting the autoimmune hypothesis of Satoyoshi syndrome, a rare multisystem disease with presumed autoimmune origins.
A thorough search was conducted in PubMed, the Web of Science, and Scopus databases up to January 2022, using the keywords “Satoyoshi syndrome” or “Komuragaeri disease.” Data pertaining to symptoms associated with autoimmune diseases, the presence of autoantibodies, and treatment response were collected and analyzed.
A total of 77 patients from 57 articles published between 1967 and 2021 were included in the study, with 59 of them being women. The mean age at diagnosis was 21.2 years—all cases presented with painful muscular spasms and alopecia. Frequent manifestations included diarrhea, malabsorption, growth retardation, amenorrhea, and bone deformities. Satoyoshi syndrome was found to be associated with other autoimmune diseases such as myasthenia gravis, autoimmune thyroiditis, idiopathic thrombocytopenic purpura, atopic dermatitis, bronchial asthma, and lupus erythematosus. Autoantibody determinations were conducted in 39 patients, of which 27 showed positive results. The most commonly detected autoantibodies were ANAs (antinuclear antibodies). Other less frequently found autoantibodies included anti-acetylcholine receptor antibodies, anti-DNA antibodies, antithyroid antibodies, anti-glutamic acid decarboxylase (anti-GAD) antibodies, and anti-gliadin antibodies. Pharmacological treatment was reported in 50 patients, and most showed improvement with corticosteroids (CS), immunosuppressants, immunoglobulins, or a combination of these medications.
Satoyoshi syndrome appears to be linked to other autoimmune diseases and is associated with various autoantibodies. Around 90% of cases showed improvement after treatment with CS or other immunosuppressants, further supporting the autoimmune hypothesis for Satoyoshi syndrome. Additional studies, including systematic determination of autoantibodies in all patients with Satoyoshi syndrome, were needed to enhance the understanding of the rare disease.
Source: academic.oup.com/rheumatology/article-abstract/62/7/2343/7030688?redirectedFrom=fulltext