A young female patient suffering from Satoyoshi syndrome had the first characteristic signs and symptoms of hair loss and progressive spontaneous intermittent painful spasms of limb muscles at age 6.5 years. Thereafter, she developed chronic diarrhea, amenorrhea, and skeletal deformities. In the orofacial region, she suffered from painful spasms of the masseter (jaw closing) muscles, progressive tooth loss, and degeneration of the mandibular condyles. Treatment with steroids and provision of complete dentures improved the signs and symptoms. Early diagnosis and timely provision of multidisciplinary care can minimize complications in these patients and improve their orofacial functions and quality of life.
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Impending central retinal vein occlusion and granulomatous uveitis in a patient with Satoyoshi syndrome.
Medicine (Baltimore)
March 2023
Department of Ophthalmology, Jichi Medical University Saitama Medical Center, Saitama, Japan.
Rationale: Satoyoshi syndrome is a rare systemic autoimmune disease that presents with painful muscle spasms. We report a case of impending central retinal vein occlusion (CRVO) secondary to granulomatous pan-uveitis in a 32-year-old woman with Satoyoshi syndrome.
Patient Concerns: At the age of 7 years, she developed generalized hair loss and painful spasms and was diagnosed with Satoyoshi syndrome.
Is Satoyoshi syndrome an autoimmune disease? A systematic review.
Rheumatology (Oxford)
July 2023
Department of Internal Medicine, Complejo Hospitalario Universitario de Albacete, Albacete, Spain.
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'.
[A case of adult-onset Satoyoshi syndrome with monoclonal gammopathy of undetermined significance].
Zhonghua Nei Ke Za Zhi
January 2023
Department of Neurology, Peking University Third Hospital, Beijing 100191, China.
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Neurology Unit, Kedah Medical Centre, Alor Setar, Kedah, Malaysia.
Adenocarcinoma arising in the multiple heterotopic submucosal glands of the intestine in a Satoyoshi syndrome patient: A case report.
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Department of Comprehensive Pathology, Tokyo Medical and Dental University Graduate School of Medical and Dental Sciences, Tokyo, Japan.
Satoyoshi syndrome is a rare multisystemic disorder of unknown etiology characterized by progressive muscle spasms, alopecia and diarrhea. Multiple protruding lesions with cystic glands, namely gastroenterocolitis cystica polyposa, manifest in the gastrointestinal tract. Since the first report of these lesions in 1977, which was unique to Satoyoshi syndrome, few studies have focused on their role, and the associated clinicopathological features are not well understood.
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