Alopecia areata. How not to miss Satoyoshi syndrome?
Lidia Rudnicka
Department of Dermatology, Medical University of Warsaw, Warsaw, Poland
Department of Neuropeptides, Mossakowski Medical Research Centre, Polish Academy of Sciences, Warsaw, Poland
Search for more papers by this authorMałgorzata Kwiatkowska
Department of Dermatology, CSK MSW, Warsaw, Poland
Search for more papers by this authorAdriana Rakowska
Department of Dermatology, Medical University of Warsaw, Warsaw, Poland
Search for more papers by this authorJoanna Czuwara
Department of Dermatology, Medical University of Warsaw, Warsaw, Poland
Search for more papers by this authorCorresponding Author
Małgorzata Olszewska
Department of Dermatology, Medical University of Warsaw, Warsaw, Poland
Correspondence: Malgorzata Olszewska, M.D., Ph.D., Department of Dermatology, Medical University of Warsaw, Koszykowa 82A, 02-008 Warsaw, Poland. Email: [email protected]Search for more papers by this authorLidia Rudnicka
Department of Dermatology, Medical University of Warsaw, Warsaw, Poland
Department of Neuropeptides, Mossakowski Medical Research Centre, Polish Academy of Sciences, Warsaw, Poland
Search for more papers by this authorMałgorzata Kwiatkowska
Department of Dermatology, CSK MSW, Warsaw, Poland
Search for more papers by this authorAdriana Rakowska
Department of Dermatology, Medical University of Warsaw, Warsaw, Poland
Search for more papers by this authorJoanna Czuwara
Department of Dermatology, Medical University of Warsaw, Warsaw, Poland
Search for more papers by this authorCorresponding Author
Małgorzata Olszewska
Department of Dermatology, Medical University of Warsaw, Warsaw, Poland
Correspondence: Malgorzata Olszewska, M.D., Ph.D., Department of Dermatology, Medical University of Warsaw, Koszykowa 82A, 02-008 Warsaw, Poland. Email: [email protected]Search for more papers by this authorAbstract
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.
References
- 1Merino de Paz N, Rodriguez-Martin M, Contreras Ferrer P, Eliche MP, Noda Cabrera A. Satoyoshi syndrome: a cause of alopecia universalis in association with neurologic and bony abnormalities. Pediatr Dermatol 2013; 30: 22–24.
- 2Mukhopadhyay D, Ghosh A, Mukhopadhyay M. Satoyoshi syndrome. Indian Pediatr 2011; 48: 729–731.
- 3Satoyoshi E. Satoyoshi syndrome. Brain Nerve 2011; 63: 141–146.
- 4Satoyoshi E. Recurrent muscle spasms of central origin. Trans Am Neurol Assoc 1967; 92: 153–157.
- 5Merello M, Garcia H, Nogues M, Leiguarda R. Masticatory muscle spasm in a non-Japanese patient with Satoyoshi syndrome successfully treated with botulinum toxin. Mov Disord 1994; 9: 104–105.
- 6Cecchin CR, Felix TM, Magalhaes RB, Furlanetto TW. Satoyoshi syndrome in a Caucasian girl improved with glucocorticoids – a clinical report. Am J Med Genet A 2003; 118A: 52–54.
- 7Castiglioni C, Diaz A, Moenne K, Mericq V, Salvador F, Hernandez C. Satoyoshi syndrome: report of one case. Rev Med Chil 2009; 137: 542–546.
- 8Li J, Jiang T, Feng HL. [A case report of dental abnormality and prosthetic treatment of Satoyoshi syndrome]. Zhonghua Kou Qiang Yi Xue Za Zhi 2008; 43: 213–215.
- 9Heger S, Kuester RM, Volk R, Stephani U, Sippell WG. Satoyoshi syndrome: a rare multisystemic disorder requiring systemic and symptomatic treatment. Brain Dev 2006; 28: 300–304.
- 10Drost G, Verrips A, van Engelen BG, Stegeman DF, Zwarts MJ. Involuntary painful muscle contractions in Satoyoshi syndrome: a surface electromyographic study. Mov Disord 2006; 21: 2015–2018.
- 11Drost G, Verrips A, Hooijkaas H, Zwarts M. Glutamic acid decarboxylase antibodies in Satoyoshi syndrome. Ann Neurol 2004; 55: 450–451.
- 12Haymon M, Willis RB, Ehlayel MS, Lacassie Y. Radiological and orthopedic abnormalities in Satoyoshi syndrome. Pediatr Radiol 1997; 27: 415–418.
- 13Satoyoshi E. Contribution of Japanese researchers to progress in the field of neurology in the last 100 years: Satoyoshi syndrome. Nihon Naika Gakkai Zasshi 2002; 91: 2325–2328.
- 14Venegas-Vega CA, Rivera-Vega MR, Cuevas-Covarrubias S, Orozco J, Kofman-Alfaro S. Satoyoshi syndrome with unusual skeletal abnormalities and parental consanguinity. Am J Med Genet A 2009; 149A: 2448–2451.
- 15Aver'ianov Iu N, Vodolagin VD, Logunova LV, Levina L. Positive therapeutic effect of diacarb in the syndrome of progressive muscle spasms, alopecia and diarrhea (Satoyoshi syndrome). Zh Nevropatol Psikhiatr Im S S Korsakova 1984; 84: 1623–1627.
- 16Asherson RA, Giampaolo D, Strimling M. A case of adult-onset Satoyoshi syndrome with gastric ulceration and eosinophilic enteritis. Nat Clin Pract Rheumatol 2008; 4: 439–444.
- 17Pardal-Fernandez JM, Solera-Santos J, Iniesta-Lopez I, Rodriguez-Vazquez M. Satoyoshi's syndrome related muscle spasms: functional study. Rev Neurol (Paris) 2012; 168: 291–295.
- 18Wisuthsarewong W, Likitmaskul S, Manonukul J. Satoyoshi syndrome. Pediatr Dermatol 2001; 18: 406–410.
- 19Kamat D, Petry L, Berry S. A case of Satoyoshi syndrome: a multisystem disorder. Clin Pediatr 2003; 42: 745–748.
- 20Dhamija R, Renaud DL, Pittock SJ et al. Neuronal voltage-gated potassium channel complex autoimmunity in children. Pediatr Neurol 2011; 44: 275–281.
- 21Uddin AB, Walters AS, Ali A, Brannan T. A unilateral presentation of ‘Satoyoshi syndrome’. Parkinsonism Relat Disord 2002; 8: 211–213.
- 22Oyama M, Imaizumi T, Mitsuhashi Y, Kondo S. Satoyoshi syndrome. Arch Dermatol 1999; 135: 91–92.
- 23Yamagata T, Miyao M, Momoi M, Matsumoto S, Yanagisawa M. A case of generalized komuragaeri disease (Satoyoshi disease) treated with glucocorticoid. Rinsho Shinkeigaku 1991; 31: 79–83.
- 24Yoshikawa K, Katagata Y, Kondo S. Relative amounts of keratin 17 are higher than those of keratin 16 in hair-follicle-derived tumors in comparison with nonfollicular epithelial skin tumors. J Invest Dermatol 1995; 104: 396–400.
- 25Arita J, Hamano S, Nara T, Maekawa K. Intravenous gammaglobulin therapy of Satoyoshi syndrome. Brain Dev 1996; 18: 409–411.
- 26Satoh A, Tsujihata M, Yoshimura T, Mori M, Nagataki S. Myasthenia gravis associated with Satoyoshi syndrome: muscle cramps, alopecia, and diarrhea. Neurology 1983; 33: 1209–1211.
- 27Wang DX, Fu HD. Three cases of recurrent generalized muscle spasms in China. Jpn J Med 1985; 24: 263–268.
- 28Stephani UVR, Steen A, Waltz S, Oppermann H-C. Satoyoshi's syndrome in two German sisters. Eur J Paediatr Neurol 1999; 3: A67.
10.1016/S1090-3798(99)91165-0 Google Scholar
- 29Ikeda K, Satoyoshi E, Kinoshita M, Wakata N, Iwasaki Y. Satoyoshi's syndrome in an adult: a review of the literature of adult onset cases. Intern Med 1998; 37: 784–787.
- 30Satoyoshi E. Satoyoshi's syndrome: a syndrome of progressive muscle spasm, alopecia, and diarrhea. Nihon Rinsho 1990; 48: 1540–1546.
- 31Sunohara N. Satoyoshi syndrome (disease). Ryoikibetsu Shokogun Shirizu 2001; 36: 384–388.
- 32Ikegawa S, Nagano A, Nakamura K, Kurokawa T. A case of Satoyoshi's syndrome. J Pediatr Orthop 1993; 13: 793–796.
- 33Matsumura T, Yokoe M, Shinno S. A case of Satoyoshi syndrome complicating marginal gingivitis of the mandible and dislocation of the temporomandibular joint. Rinsho Shinkeigaku 2002; 42: 889–891.
- 34Nagahama T, Yao K, Oishi T et al. GI manifestations of Satoyoshi's syndrome. Gastrointest Endosc 2006; 64: 143–145.
- 35Matsuura E, Matsuyama W, Sameshima T, Arimura K. Satoyoshi syndrome has antibody against brain and gastrointestinal tissue. Muscle Nerve 2007; 36: 400–403.
- 36Ashalatha R, Kishore A, Sarada C, Nair MD. Satoyoshi syndrome. Neurol India 2004; 52: 94–95.
- 37Son KR, Kook JH, Kim BJ, Kim SJ, Ma JS. A case of Satoyoshi syndrome presented with progressive muscular spasm and alopecia. Korean J Pediatr 2002; 45: 1165–1169.
- 38Adachi H, Riku S, Fujishiro K, Kuru S. A case of Satoyoshi syndrome with symptoms resembling neuroleptic malignant syndrome. Rinsho Shinkeigaku 1998; 38: 637–640.
- 39Kuru S, Riku S, Nakayabu M, Kobayashi Y, Ieda T. A case of “syndrome of progressive muscle spasm, alopecia, and diarrhea (Satoyoshi)” treated with steroid pulse therapy. Rinsho Shinkeigaku 1992; 32: 612–615.
- 40Satoyoshi E. Establishment of a syndrome of progressive muscle spasm, alopecia and diarrhea (author's transl). Rinsho Shinkeigaku 1978; 18: 731–739.
- 41Matsuo N, Fujioka M, Tsuchiya Y, Cho H, Nagai T, Kumagai M. Multiple metaphyseal lesions in a child with a syndrome of progressive muscle cramps, alopecia and stunted growth (Satoyoshi disease). Radiat Med 1983; 1: 205–207.
- 42Ikegawa S, Nagano A, Satoyoshi E. Skeletal abnormalities in Satoyoshi's syndrome: a radiographic study of eight cases. Skeletal Radiol 1993; 22: 321–324.
- 43Ehlayel MS, Lacassie Y. Satoyoshi syndrome: an unusual postnatal multisystemic disorder. Am J Med Genet 1995; 57: 620–625.
- 44Furue M, Yamazaki S, Jimbow K et al. Prevalence of dermatological disorders in Japan: a nationwide, cross-sectional, seasonal, multicenter, hospital-based study. J Dermatol 2011; 38: 310–320.
- 45Bilgili ME, Yildiz H, Sarici G. Prevalence of skin diseases in a dermatology outpatient clinic in Turkey. A cross-sectional, retrospective study. J Dermatol Case Rep 2013; 7: 108–112.
- 46Inui S. Trichoscopy for common hair loss diseases: algorithmic method for diagnosis. J Dermatol 2011; 38: 71–75.
- 47Ise M, Amagai M, Ohyama M. Follicular microhemorrhage: a unique dermoscopic sign for the detection of coexisting trichotillomania in alopecia areata. J Dermatol 2014; 41: 518–520.
- 48Lencastre A, Tosti A. Role of trichoscopy in children's scalp and hair disorders. Pediatr Dermatol 2013; 30: 674–682.
- 49Rudnicka L, Olszewska M, Rakowska A, Kowalska-Oledzka E, Slowinska M. Trichoscopy: a new method for diagnosing hair loss. J Drugs Dermatol 2008; 7: 651–654.