Julián Solís García del Pozo

Background/Objectives: Satoyoshi syndrome is a rare, autoimmune disorder currently diagnosed based on clinical criteria: painful muscle spasms, diarrhea, and alopecia. Two previous reports showed a specific immunoreactive band in three Satoyoshi syndrome patients using Western blot analysis, with brain homogenate as the antigen source. These findings could be the basis for a future diagnostic test. The aim of our study was to evaluate the efficacy of using SH-SY5Y cell lysate instead of brain homogenate for a potential laboratory test for Satoyoshi syndrome using the Western blot technique. Methods: Western blot analyses were conducted using brain homogenate, SH-SY5Y cell lysates, and differentiated SH-SY5Y cell lysates. Serum samples were obtained from three Satoyoshi syndrome patients, alongside control samples from thirty blood donors and six patients with other neurological conditions. Results: Sera from patients with Satoyoshi syndrome displayed a three-band pattern in the 70–100 kDa range. This pattern was reproducible across all tested antigen sources (brain homogenate, SH-SY5Y lysate, and differentiated SH-SY5Y lysate) but was not observed for the sera from the control groups. The bands were more visible when using either type of SH-SY5Y lysate compared to brain homogenate. No differences were found between the SH-SY5Y lysate and the differentiated SH-SY5Y lysate. Conclusions: Sera from our Satoyoshi syndrome patients showed a specific band pattern that could be used for a future evaluation of Satoyoshi syndrome using Western blot. The use of SH-SY5Y cell lysate vs. brain homogenate as an antigen source may improve visualization and reproducibility of the immunobands and be less costly.

Objective: Satoyoshi syndrome is a rare, likely autoimmune disorder that is traditionally diagnosed based on clinical criteria: painful muscle spasms, diarrhea and alopecia. Two previous reports showed a specific immunoreactive band in three patients using Western blot analysis with brain homogenate. The aim of this study was to evaluate the efficacy of SH-SY5Y cell lysate as a potential substitute for brain homogenate in the diagnosis of Satoyoshi syndrome. Methods: Western blot analyses were conducted using brain homogenate, SH-SY5Y cell lysates, and differentiated SH-SY5Y cell lysates. Serum samples were obtained from three patients diagnosed with Satoyoshi syndrome, alongside control samples from thirty blood donors and six patients with other neurological conditions. Results: Sera from patients with Satoyoshi syndrome displayed a consistent three band pattern in the 70–100 kDa range. This pattern was reproducible across all tested substrates (brain homogenate, SH-SY5Y lysate, and differentiated SH-SY5Y lysate), but it was not observed for the sera from the control groups. The immunoreactive bands were more patent when using either SH-SY5Y lysate compared to brain homogenate. No differences were found between the SH-SY5Y lysate and differentiated SH-SY5Y lysate. Conclusion: SH-SY5Y cell lysate could be an alternative to brain homogenate for the immunodiagnostic evaluation of Satoyoshi syndrome. The use of SH-SY5Y cell lysate for Western blot analysis may improve visualization and reproducibility and have a lower cost. Key messages: Satoyoshi syndrome is a rare disease currently diagnosed based only on clinical criteria A biological marker test for Satoyoshi syndrome could lead to early diagnosis and treatment thus improving the outcomes. SH-SY5Y cell lysate may be a better substrate than brain homogenate for Western blot based diagnosis of Satoyoshi syndrome.

Background: This work aims to establish the relationship between depression and epidemiological or imaging variables, frailty, and cognitive status in patients who suffered hospital admission for COVID-19. Methods: A longitudinal observational study investigated 72 patients admitted for COVID-19 to a hospital in Spain. Patients were evaluated at discharge and six months later. Clinical, analytical, and imaging variables were collected. A neurocognitive, nutritional, and frailty (FRAIL scale) assessment of the included patients was carried out. The risk of depression was considered for a result above 5 points on the PHQ-9 scale. Results: The variables that were significantly related to the risk of depression 6 months after admission for COVID-19 were frailty (p = 0.006 for pre-frail and p = 0.001 for frail), small-vessel vascular disease in imaging tests (p = 0.033), vitamin D level (p = 0.006), and taking antidepressants (p = 0.011). Factors that were negatively associated with the presence of depression 6 months after discharge were a higher score on the CAMCOG cognitive scale (p = 0.041) and older age (p = 0.006). Conclusions: Frailty worsened the score on the PHQ-9 depression scale in patients who required hospital admission for SARS-CoV-2 infection. It is important to implement prevention measures both for frailty and depression in these patients.

Objectives: Satoyoshi syndrome is a rare multisystem disease of presumed autoimmune aetiology. We carried out a systematic review to evaluate the available evidence to support that autoimmune hypothesis.Methods: We searched for Satoyoshi syndrome cases in PubMed, the Web of Science and Scopus up to January 2022, using keywords ‘Satoyoshi syndrome’ or ‘Komuragaeri disease’. Data on symptoms, associated autoimmune diseases, presence of autoantibodies and response to treatment were collected.Results: A total of 77 patients from 57 articles published between 1967 and 2021 were included; 59 patients were women. The mean age at diagnosis was 21.2 years. All cases had painful muscular spasms and alopecia. Frequent manifestations included: diarrhoea, malabsorption, growth retardation, amenorrhoea and bone deformity. Satoyoshi syndrome was associated with other autoimmune diseases: myasthenia gravis, autoimmune thyroiditis, idiopathic thrombocytopenic purpura, atopic dermatitis, bronchial and lupus erythematosus. Autoantibody determinations were performed in 39 patients, of which 27 had positive results. The most frequently detected autoantibodies were ANAs. Other less frequently found autoantibodies were: anti-acetylcholine receptor antibodies, anti-DNA antibodies, antithyroid antibodies, anti-glutamic acid decarboxylase (anti-GAD) and anti-gliadin antibodies. Pharmacological treatment was reported in 50 patients. Most of them improved with CS, immunosuppressants and immunoglobulins, or a combination of these medications.Conclusion: Satoyoshi syndrome is associated with other autoimmune diseases and a variety of autoantibodies. Improvement after CS or other immunosuppressant treatment was observed in 90% of cases. These data support an autoimmune aetiology for Satoyoshi syndrome. More studies including systematic determination of autoantibodies in all patients with Satoyoshi syndrome will help us advance in our understanding of this disease.

Background Satoyoshi syndrome (SS) is a rare multisystem disease. Although its cause is unknown, an autoimmune etiology has been postulated. In this paper we carried out a systematic review of all published cases of SS to evaluate the available evidence to support that autoimmune hypothesis. Methods We carried out a systematic review of the published cases of SS following the recommendations of the PRISMA statement for systematic reviews. We searched for SS cases in PubMed, the Web of Knowledge (WOS) and Scopus up to January 2022, using keywords “Satoyoshi syndrome” or “Komuragaeri disease”. Data on symptoms, associated autoimmune diseases, presence of autoantibodies and response to treatment were collected. Results 77 patients from 57 articles published between 1967 and 2021 were included. 59 patients were women. The mean age at diagnosis was 21.2 years. All cases had painful muscular spasms and alopecia. Other frequent manifestations included: diarrhea, malabsorption, growth retardation, amenorrhea and bone deformity. SS was associated with other autoimmune diseases: myasthenia gravis (2 patients), autoimmune thyroiditis (one patient), idiopathic thrombocytopenic purpura (one patient), atopic dermatitis (one patient), bronchial asthma (one patient) and lupus erythematosus (one patient). Autoantibody determinations were performed in 39 patients, of which 27 had positive results. The most frequently detected autoantibodies were antinuclear antibodies (21 patients). Other less frequently found auto-antibodies were: anti-acetylcholine receptor antibodies (7 patients), anti-DNA antibodies (5 patients), antithyroid antibodies (3 patients), anti-GAD (2 patients) and anti-gliadin antibodies (2 patients). Pharmacological treatment was reported in 50 patients. Most of them improved with corticosteroids (33 patients), immunosuppressants (9 patients) and immunoglobulins (10 patients), or a combination of these medications. Conclusions SS is associated with other autoimmune diseases and a variety of autoantibodies. Improvement after corticosteroid or other immunosupressant treatment was observed in 90% of cases. These data support an autoimmune etiology for SS. More studies are necessary, including the systematic determination of autoantibodies in all patients with SS to help us advance in our understanding of this disease.