Papers - KAWAKAMI Yoshio

Development of antigen-specific ELISA for circulating autoantibodies to extracellular matrix protein 1 in lichen sclerosus. International journal

Noritaka Oyama, Ien Chan, Sallie M Neill, Andrew P South, Fenella Wojnarowska, Yoshio Kawakami, David D'Cruz, Kirti Mepani, Graham J Hughes, Balbir S Bhogal, Fumio Kaneko, Martin M Black, John A McGrath

The Journal of clinical investigation   113 ( 11 )   1550 - 9   2004.6

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Lichen sclerosus is a common, acquired chronic inflammatory skin disease of unknown etiology, although circulating autoantibodies to the glycoprotein extracellular matrix protein 1 (ECM1) have been detected in most patients' sera. We have examined the nature of ECM1 epitopes in lichen sclerosus sera, developed an ELISA system for serologic diagnosis, and assessed clinicopathological correlation between ELISA titer and disease. Epitope-mapping studies revealed that lichen sclerosus sera most frequently recognized the distal second tandem repeat domain and carboxyl-terminus of ECM1. We analyzed serum autoantibody reactivity against this immunodominant epitope in 413 individuals (95 subjects with lichen sclerosus, 161 normal control subjects, and 157 subjects with other autoimmune basement membrane or sclerosing diseases). The ELISA assay was highly sensitive; 76 of 95 lichen sclerosus patients (80.0%) exhibited IgG reactivity. It was also highly specific (93.7%) in discriminating between lichen sclerosus and other disease/control sera. Higher anti-ECM1 titers also correlated with more longstanding and refractory disease and cases complicated by squamous cell carcinoma. Furthermore, passive transfer of affinity-purified patient IgG reproduced some histologic and immunopathologic features of lichen sclerosus skin. This new ELISA is valuable for the accurate detection and quantification of anti-ECM1 autoantibodies. Moreover, the values may have clinical significance in patients with lichen sclerosus.

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A case of generalized pustular psoriasis associated with Turner syndrome. International journal

Yoshio Kawakami, Noritaka Oyama, Kazuhiro Kishimoto, Keiji Yamazaki, Akiko Nishibu, Koichiro Nakamura, Satoshi Hoshi, Fumio Kaneko

The Journal of dermatology   31 ( 1 )   16 - 20   2004.1

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UNLABELLED: We report a 27-year-old Japanese woman with Turner syndrome who had generalized pustular psoriasis of the von Zumbusch type. She developed a febrile diffuse erythema and pustular eruption without any history of preceding psoriasis vulgaris or drug ingestion. Oral treatment with 3.2 mg/kg cyclosporin per day successfully resulted in rapid improvement, followed by a complete remission. To our knowledge, this is the first report describing the unusual coexistence of these two systemic disorders. We discuss a hormone imbalance that might have contributed to the predisposition to pustular psoriasis and difficulties in the management of the patent's treatment. ABBREVIATIONS: TS: Turner syndrome, GPP: generalized pustular psoriasis

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A case of intravascular large B-cell lymphoma with multiple organ involvement. International journal

Hirokatsu Yanagihori, Noritaka Oyama, Yoshio Kawakami, Yohko Sakuma-Oyama, Koichiro Nakamura, Keiji Iwatsuki, Fumio Kaneko

The Journal of dermatology   30 ( 12 )   910 - 4   2003.12

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Intravascular large B-cell lymphoma (IVL) is a rare systemic disease characterized by clonal proliferation of neoplastic lymphoid cells within the capillary lumina of small blood vessels. The most common sites of involvement are cerebrovascular or sinusoid areas, resulting in a wide variety of neurologic deficits. Herein we describe a case of IVL that presented with multiple internal and external organ involvements, including the skin, bilateral kidneys, and lung, and with an aggressive clinical course. The confirmative diagnosis was based on the microscopic findings of the skin lesion with a B-cell immunophenotype. The patient suffered from repeated severe dyspnea, hypoxemia, and high-grade fever with a simultaneous increase in serum lactate dehydrogenase and soluble IL-2 receptor levels. These manifestations responded dramatically to intravenous injection of corticosteroid (500 mg/day for 3 days). Despite various clinical presentations, no neurological abnormality was observed until the patient died 12 months after the initial combination chemotherapy.

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Multicentric Castleman's disease associated with inherited epidermolysis bullosa. International journal

Yoshio Kawakami, Akiko Nishibu, Satoshi Kikuchi, Mikio Ohtsuka, Koichiro Nakamura, Yoshihiro Nozawa, Masafumi Abe, Keiji Iwatsuki, Fumio Kaneko

The Journal of dermatology   30 ( 9 )   689 - 93   2003.9

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Multicentric Castleman's disease (MCD) is a rare disorder characterized by fever, polyclonal hypergammaglobulinemia, and generalized lymphadenopathy. It has three histological characteristics: a recognizable architecture, germinal center abnormalities, and plasmacytosis. Inherited epidermolysis bullosa (EB) is also a rare disorder caused by a genetic defect. We report a 43-year-old patient with dystrophic EB, non-Hallopeau-Siemens recessive type or dominant type, displaying clinicopathologic features of MCD. In addition, his serum interleukin-6, which is thought to be responsible for the clinical symptoms in MCD, was elevated.

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