A Japanese Woman with Stevens-Johnson Syndrome without Skin Lesions Induced by an Initial Dosage of Lamotrigine
American Journal of Clinical and Experimental Medicine
Volume 7, Issue 1, January 2019, Pages: 7-11
Received: Dec. 16, 2018;
Accepted: Feb. 27, 2019;
Published: Mar. 19, 2019
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Yasuyuki Sumida, Emergency Department, North Medical Center Kyoto Prefectural University of Medicine, Yosanocho, Japan
Hidetaka Ishino, Department of General Internal Medicine, North Medical Center Kyoto Prefectural University of Medicine, Yosanocho, Japan
Atsushi Saitou, Department of Otolaryngology, Kyoto Second Red Cross Hospital, Kyoto, Japan
Yohei Otsuki, Department of Ophthalmology, Kyoto Prefectural University of Medicine, Kyoto, Japan
Satoshi Komori, Department of Dermatology, Kyoto Prefectural University of Medicine, Kyoto, Japan
Atsuhiro Kanzaki, Department of Psychiatry, Kyoto Prefectural University of Medicine, Kyoto, Japan
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Atypical Stevens-Johnson syndrome (SJS) which is characterized by little or no skin involment, is reported to be triggered by less medications than infection. There are few reports with atypical SJS induced by medications. This is a case report of atypical SJS induced by Lamotrigine (LTG). A 31-year-old Japanese woman, who was bipolar disorder and prescribed an initial dosage of LTG 25mg/day ten days ago, presented with high fever and throat pain for two days. At first she was suspected of pharyngitis and administered antibiotics. She was hospitalized on the first visit day, but she was getting worse and fell into shock over several days. Furthermore she presented with erosions and bleeding of her lips and oral mucosa on day6, and so she was diagnosed with SJS, but her skin lesions were mild even at the extreme of the clinical course. She gradually developed acute pulmonary failure, liver dysfunction, coma and Disseminated Intravascular Coagulation (DIC). But she was treated with intensive care consisting of steroid pulse followed by high dose corticosteroids, and so she gradually recovered from SJS and corticosteroids could be tapered. At last she was discharged on day50 and had no sequelae with SJS. It was extremely difficult to diagnose this case as atypical SJS because of the unremarkable skin lesions and similarity to pharyngitis. It was also characteristic that she was already in severe status when it satisfied the diagnostic criteria for SJS. But physicians should not miss from the perspective of the fatality and social problem. It should be noted that atypical SJS shows mild or little cutaneous lesions.
Atypical, Incomplete, Fuchs Syndrome, Lamotrigine, Stevens-Johnson Syndrome, Without Skin Lesions, Without Skin Involvement
To cite this article
A Japanese Woman with Stevens-Johnson Syndrome without Skin Lesions Induced by an Initial Dosage of Lamotrigine, American Journal of Clinical and Experimental Medicine.
Vol. 7, No. 1,
2019, pp. 7-11.
Copyright © 2019 Authors retain the copyright of this article.
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Michiko Aihara, Yoko Kano and Masahumi Iijima et al (2009). Guidelines for the Management of Stevens-Johnson Syndrome and Toxic Epidermal Necrolysis 2009, Established by the Japanese Research Committee on Severe Adverse Reaction (J-SCAR) Supported by the Ministry of Health, Labour and Welfare of Japan. Japanese Journal of Dermatology 119, 2157-63 (in Japanese).
Havliza K, Jakob A and Rompel R (2009). Erythema multiforme majus (Fuchs syndrome) associated with mycoplasma pneumonia (Stevens-Johnson syndrome without skin lesions). J Dtsch Dermatol Ges 7, 445-7.
Meyer Sauteur PM, Gansser-Kälin U and Lautenschlager S et al (2011). Fuchs syndrome associated with mycoplasma pneumoniae (Stevens–Johnson syndrome without skin lesions). Pediatr Dermatol 28, 474–6.
Thomas M. Nappe, Stephanie L. Goren-Garcia, and Jeanne L. Jacoby (2016). Stevens-Johnson syndrome after treatment with azithromycin: an uncommon culprit. Am J Emerg Med 34, 676.e1-3.
Shailendra Kapoor (2016). Fuchs syndrome: A rare and unique variant of Stevens-Johnson Syndrome. Am J Emerg Med 34, 910.
Kayi Li and Richard M. Haber (2012). Stevens-Johnson syndrome without skin lesions (Fuchs syndrome): a literature review of adult cases with mycoplasma cause. Arch Dermatol 148, 963-4.
Patrick M. Meyer Sauteur, Philippe Goetschel and Stephan Lautenschlager (2012). Mycoplasma pneumoniae and mucositis–part of the Stevens-Johnson syndrome spectrum. J Dtsch Dermatol Ges 10, 740-6.
Peter C. Schalock, and James G. H. Dinulos (2005). Mycoplasma pneumoniae-induced Stevens-Johnson syndrome without skin lesions: fact or fiction?. J Am Acad Dermatol 52, 312-5.
Rémy Gossart, Eve Malthiery, Fanny Aguilar, Jacques-Henri Torres, and Marie-Alix Fauroux (2017). Fuchs Syndrome: Medical Treatment of 1 Case and Literature Review. Case Rep Dermatol 9, 114–20.
Chun Yi Ting and Raveen Shahdadpuri (2018). Fuchs syndrome: a case of fever, mucositis and conjunctivitis. BMJ Case Rep 2018 Jan 29.
Reyhan Amode, Saskia Ingen-Housz-Oro, and Nicolas Ortonne et al (2018). Clinical and histologic features of Mycoplasma pneumoniae–related erythema multiforme: A single-center series of 33 cases compared with 100 cases induced by other causes. J Am Acad Dermatol 79, 110-7.
Canavan TN, Mathes EF, and Frieden I et al (2015). Mycoplasma pneumoniae-induced rash and mucositis as a syndrome distinct from Stevens-Johnson syndrome and erythema multiforme: A systematic review. J Am Acad Dermatol 72, 239-45.
I. Vujic, A. Shroff and M. Grzelka et al (2015). Mycoplasma pneumoniae-associated mucositis-case report and systematic review of literature. J Eur Acad Dermatol Venereol 29, 595-8.
David A. Wetter and Michael J. Camilleri (2009). Clinical, Etiologic, and Histopathologic Features of Stevens-Johnson Syndrome During an 8-Year Period at Mayo Clinic. Mayo Clin Proc 85, 131-138.
Kunimi Y, Hirata Y, and Aihara M et al (2011). Statistical Analysis of Stevens-Johnson Syndrome Caused by Mycoplasma pneumonia Infection in Japan. Allergol Int 60, 525-32.
Romi Bloom and Kyle T. Amber (2017). Identifying the incidence of rash, Stevens-Johnson syndrome and toxic epidermal necrolysis in patients taking lamotrigine: a systematic review of 122 randomized controlled trials. An Bras Dermatol 92, 139-41.