396 Satoyoshi's syndrome in two German sisters

Stephani, Ulrich; Volk, Richard; Steen, Andreas Van; Waltz, S.; Oppermann, H

doi:10.1016/s1090-3798(99)91165-0

Cited by 3 publications

(5 citation statements)

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“…Growth retardation was observed in 74.1% of patients with an onset of disease before the age of 18 and in none of the adult patients . Amenorrhea was reported in 50% of adult women.…”

Section: Diarrhea and Malabsorptionmentioning

confidence: 95%

“…Amenorrhea was reported in 50% of adult women. This was secondary amenorrhea in patients with adult disease onset and primary amenorrhea in women who had the first symptoms of disease before puberty …”

Section: Diarrhea and Malabsorptionmentioning

confidence: 99%

“…Growth retardation was observed in 74.1% of patients with an onset of disease before the age of 18 and in none of the adult patients. 1,6,19,28,[34][35][36] Amenorrhea was reported in 50% of adult women. This was secondary amenorrhea in patients with adult disease onset 22,34 and primary amenorrhea in women who had the first symptoms of disease before puberty.…”

Section: Diarrhea and Malabsorptionmentioning

confidence: 99%

“…Family history is negative in patients with Satoyoshi syndrome, which may indirectly indicate that there is no significant genetic basis for the disorder. There was only one conference communication of Satoyoshi syndrome in two sisters and one case of Satoyoshi syndrome in a child of consanguineous parents …”

Section: Pathogenesismentioning

confidence: 99%

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Alopecia areata. How not to miss Satoyoshi syndrome?

Rudnicka

Kwiatkowska

Rakowska

et al. 2014

The Journal of Dermatology

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Satoyoshi syndrome is a multisystem disorder of suspected autoimmune etiology, characterized predominantly by alopecia, muscle spasms and diarrhea. Antinuclear antibodies are present in 60% of patients. The syndrome primarily affects girls and young women. Trichoscopy shows regularly distributed yellow dots, indistinguishable from typical alopecia areata. The condition may be easily misdiagnosed and treated as alopecia areata. On the basis of an in-depth analysis of all published cases we developed diagnostic criteria for Satoyoshi syndrome. We also suggest that two subtypes of the disorder should be distinguished, the ANA-positive Satoyoshi syndrome with generally good response to systemic glucocorticosteroid therapy and the ANA-negative Satoyoshi with less favorable prognosis. In our opinion all patients will alopecia areata (in particular alopecia totalis) should be inquired about muscle spasms and diarrhea and tested for antinuclear antibodies to decrease the risk of missing Satoyoshi syndrome.

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“…Growth retardation was observed in 74.1% of patients with an onset of disease before the age of 18 and in none of the adult patients . Amenorrhea was reported in 50% of adult women.…”

Section: Diarrhea and Malabsorptionmentioning

confidence: 95%

Section: Diarrhea and Malabsorptionmentioning

confidence: 99%

Section: Diarrhea and Malabsorptionmentioning

confidence: 99%

Section: Pathogenesismentioning

confidence: 99%

See 2 more Smart Citations

Alopecia areata. How not to miss Satoyoshi syndrome?

Rudnicka

Kwiatkowska

Rakowska

et al. 2014

The Journal of Dermatology

View full text Add to dashboard Cite

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“…A total of 77 cases of Satoyoshi syndrome were identified from 57 records [3-7, 9-60] and 2 personal communications. 14 records provided additional information on cases already reported in other references [61][62][63][64][65][66][67][68][69][70][71][72][73][74]. 13 were records not found [75][76][77][78][79][80][81][82][83][84][85][86][87] (Fig.…”

Section: Resultsmentioning

confidence: 99%

Autoimmunity in Satoyoshi Disease: a Systematic Review

Montanaro

Pozo

Hora

et al. 2022

Preprint

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BackgroundSatoyoshi 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.MethodsWe 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.Results77 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.ConclusionsSS 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.

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Is Satoyoshi syndrome an autoimmune disease? A systematic review

et al. 2023

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Objectives Satoyoshi syndrome (SS) is a rare multisystem disease of presumed autoimmunea aetiology. We carried out a systematic review to evaluate the available evidence to support that autoimmune hypothesis. Methods We searched for SS cases in PubMed, the Web of Knowledge 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. Frequent manifestations included: diarrhoea, malabsorption, growth retardation, amenorrhea and bone deformity. SS 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 antinuclear antibodies. Other less frequently found auto-antibodies were: anti-acetylcholine receptor antibodies, anti-DNA antibodies, antithyroid antibodies, anti-GAD and anti-gliadin antibodies. Pharmacological treatment was reported in 50 patients. Most of them improved with corticosteroids, immunosuppressants and immunoglobulins, or a combination of these medications. Conclusion SS is associated with other autoimmune diseases and a variety of autoantibodies. Improvement after corticosteroid or other immunosuppressant treatment was observed in 90% of cases. These data support an autoimmune aetiology for SS. More studies including systematic determination of autoantibodies in all patients with SS will help us advance in our understanding of this disease.

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396 Satoyoshi's syndrome in two German sisters

Cited by 3 publications

References 0 publications

Alopecia areata. How not to miss Satoyoshi syndrome?

Alopecia areata. How not to miss Satoyoshi syndrome?

Autoimmunity in Satoyoshi Disease: a Systematic Review

Is Satoyoshi syndrome an autoimmune disease? A systematic review

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