Publications by authors named "Sora Takeuchi"

30 Publications

  • Page 1 of 1

Elevated Myeloperoxidase-DNA Complex Levels in Sera of Patients with IgA Vasculitis.

Pathobiology 2021 Nov 23:1-6. Epub 2021 Nov 23.

Department of Dermatology, St. Marianna University School of Medicine, Kawasaki, Japan.

Introduction: IgA vasculitis is a systemic disease that results from the entrapment of circulating IgA-containing immune complexes in small-vessel walls in the skin, kidneys, and gastrointestinal tract. An excessive formation of neutrophil extracellular traps (NETs) is involved in the pathogenesis of vasculitis, especially in antineutrophil cytoplasmic antibody (ANCA)-associated vasculitis. This study aimed to clarify whether NETs are implicated in IgA vasculitis.

Methods: Twenty-two patients with IgA vasculitis and 4 healthy volunteers were enrolled in this study. Serum levels of myeloperoxidase (MPO)-DNA complex, a fragment derived from NETs, were determined by enzyme-linked immunosorbent assay (ELISA), and the association between MPO-DNA complex levels and clinical parameters was examined. The presence of the ANCA was also assessed by ELISA specific for MPO and proteinase 3 (PR3) and indirect immunofluorescence (IIF), followed by assessing the differences in clinical parameters with and without the ANCA.

Results: Serum MPO-DNA complex levels were significantly higher in patients with IgA vasculitis than those in healthy controls. A significant positive correlation between the serum MPO-DNA complex and IgA levels was noted. Interestingly, 63.6% of IgA vasculitis patients were ANCA-positive in IIF with an atypical pattern, whereas neither MPO-ANCA nor PR3-ANCA was detected by ELISA. These findings indicated that some IgA vasculitis patients possessed the so called minor ANCA. Serum IgA and MPO-DNA complex levels and the frequency of hematuria in the minor ANCA-positive group were significantly higher than in the minor ANCA-negative group.

Conclusion: The collective findings suggested that NETs are certainly involved in the pathogenesis of IgA vasculitis.
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http://dx.doi.org/10.1159/000519869DOI Listing
November 2021

Sitagliptin-associated bullous pemphigoid with autoantibodies against BP230 and laminin-332.

Int J Dermatol 2021 Jul 1. Epub 2021 Jul 1.

Department of Dermatology, St. Marianna University School of Medicine, Kawasaki, Kanagawa, Japan.

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http://dx.doi.org/10.1111/ijd.15762DOI Listing
July 2021

Stewart-Treves syndrome associated with disuse edema in amyotrophic lateral sclerosis.

J Dermatol 2021 Sep 31;48(9):e443-e444. Epub 2021 May 31.

Department of Dermatology, St. Marianna University School of Medicine, Kawasaki, Japan.

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http://dx.doi.org/10.1111/1346-8138.16004DOI Listing
September 2021

Complete pruritus relief by oren-gedoku-to in eruptive pruritic papular porokeratosis.

J Dermatol 2021 Aug 13;48(8):e378-e379. Epub 2021 May 13.

Department of Dermatology, St. Marianna University School of Medicine, Kawasaki, Kanagawa, Japan.

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http://dx.doi.org/10.1111/1346-8138.15947DOI Listing
August 2021

Arthritis and enthesitis during dupilumab therapy completely remitted by celecoxib.

J Dermatol 2021 Jun 28;48(6):e279-e280. Epub 2021 Mar 28.

Department of Dermatology, St Marianna University School of Medicine, Kawasaki, Japan.

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http://dx.doi.org/10.1111/1346-8138.15872DOI Listing
June 2021

A case of facial redness in atopic dermatitis occurring during dupilumab treatment successfully treated with topical delgocitinib ointment.

Dermatol Ther 2021 03 25;34(2):e14888. Epub 2021 Feb 25.

Department of Dermatology, St. Marianna University School of Medicine, Kawasaki, Kanagawa, Japan.

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http://dx.doi.org/10.1111/dth.14888DOI Listing
March 2021

Intravascular large B-cell lymphoma mimicking erythema nodosum: A case report and review of published works.

J Dermatol 2021 Apr 7;48(4):e184-e185. Epub 2021 Feb 7.

Department of Dermatology, St Marianna University School of Medicine, Kawasaki, Japan.

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http://dx.doi.org/10.1111/1346-8138.15792DOI Listing
April 2021

Atrophic erythema in a patient with immunoglobulin G4-related sclerosing sialadenitis.

J Dermatol 2020 Oct 19;47(10):e360-e362. Epub 2020 Jul 19.

Department of Dermatology, St Marianna University School of Medicine, Kawasaki, Japan.

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http://dx.doi.org/10.1111/1346-8138.15518DOI Listing
October 2020

Increase of lymphocytes and eosinophils, and decrease of neutrophils at an early stage of anti-PD-1 antibody treatment is a favorable sign for advanced malignant melanoma.

Drug Discov Ther 2020 Jul 27;14(3):117-121. Epub 2020 Jun 27.

Department of Dermatology, St. Marianna University School of Medicine, Kawasaki, Kanagawa, Japan.

The advent of immune checkpoint inhibitors such as anti-PD-1 antibodies had a striking impact on the treatment for advanced malignant melanoma. However, less than half of the patients benefited from those antibodies, and biomarkers that could sensitively differentiate responders from non-responders are urgently needed. Herein, we explored such biomarkers by retrospectively analyzing clinical data from patients with advanced malignant melanoma treated with nivolumab and pembrolizumab. We found that anti-PD-1 antibody was especially effective for those with metastasis only to soft tissues. Although no significant difference was found in the baseline value of relative neutrophil count (RNC), relative lymphocyte count (RLC), neutrophil to lymphocyte ratio (NLR), and relative eosinophil count (REC) between responders and non-responders, responders after anti-PD-1 therapy revealed the increase of lymphocytes and eosinophils and the decrease of neutrophils within the first 6 weeks of the treatment. We also calculated the change of RNC and RLC 3 weeks and 6 weeks after the initiation of the therapy and designated as NΔ3-LΔ3 and NΔ6-LΔ6 respectively. NΔ3-LΔ3 was significantly decreased in responders, which suggest that the neutrophil decrease and lymphocyte increase after as early as 3 weeks of anti-PD-1 therapy might be a useful clinical indicator. In addition, the difference of NΔ6-LΔ6 between responders and non-responders was even more robust. These data suggest that change of RNC, RLC, and REC together with the combination of NΔ3-LΔ3 and NΔ6-LΔ6 might be a useful tool for early and sensitive biomarkers for anti-PD-1 therapy.
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http://dx.doi.org/10.5582/ddt.2020.03043DOI Listing
July 2020

Relationship between lysosomal-associated membrane protein-2 and anti-phosphatidylserine/prothrombin complex antibody in the pathogenesis of cutaneous vasculitis.

Clin Exp Rheumatol 2020 Mar-Apr;38 Suppl 124(2):161-165. Epub 2020 Jan 27.

Department of Medical Laboratory Science, Faculty of Health Sciences, Hokkaido University, Sapporo, Japan.

Objectives: We investigated the relationship between lysosomal-associated membrane protein-2 (LAMP-2) and anti-phosphatidylserine/prothrombin (PS/PT) antibody in the pathogenesis of cutaneous vasculitis.

Methods: Cell surface LAMP-2 expression of human neutrophils was measured using flow cytometry. Twenty inbred wild-type Wistar-King-Aptekman-Hokudai (WKAH) rats were divided into four groups: Group 1, rabbit IgG injection only as negative control (n=5); Group 2, both histone and rabbit IgG injection (n=5); Group 3, anti-LAMP-2 antibody injection only (n=5); and Group 4, both histone and anti-LAMP-2 antibody injection (n=5). Ten WKAH rats were divided into two groups: Group A, histone, anti-PS/PT antibody, and anti-LAMP-2 antibody injection (n=5), and Group B, histone, anti-PS/PT antibody, and rabbit IgG injection as control (n=5).

Results: LAMP-2 expression on human neutrophils was induced by cell-free histone exposure in a dose- and time-dependent manner. Histopathological examination revealed the recruitment of neutrophils in cutaneous small vessels in all Group 4 rats. These observations were not evident in systemic organs other than the skin. LAMP-2 expression on the surface of vascular endothelial cells was evident in Group 2, exclusively in the skin, but not in Group 1. Thrombi were detected in various organs in all Groups A and B rats. However, no apparent thrombi were observed in the skin.

Conclusions: Anti-PS/PT and anti-LAMP-2 antibodies are responsible for independent effector mechanisms in the rats given intravenous injection of cell-free histones. We considered that undetermined factors other than cell-free histones could be required for the induction of cutaneous vasculitis by anti-PS/PT and anti-LAMP-2 antibodies.
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September 2020

Sudden elevation of plasma D-dimer levels induced by the combination therapy of dabrafenib and trametinib: Report of two cases.

J Dermatol 2019 Apr 5;46(4):358-360. Epub 2019 Feb 5.

Department of Dermatology, St Marianna University School of Medicine, Kawasaki, Japan.

The combination therapy of dabrafenib and trametinib revolutionized the treatment for BRAF V600-mutated melanoma. Various adverse events have been reported for this treatment, most notably fever. Herein, we report two cases of novel an adverse event, namely sudden and significant elevation of plasma D-dimer level induced by this therapy. In the first case, the remarkable elevation of plasma D-dimer level up to 87.4 mg/dL was noted on day 11, and in the second case, the plasma D-dimer level reached 125.5 mg/dL on day 25. In both cases, D-dimer levels gradually decreased after the cessation of this therapy. Although the exact cause is not clear, we assume two possible hypotheses: the first is that the combination therapy may induce disseminated intravascular coagulation, and the second is that the therapy induced pathological condition of secondary thrombotic microangiopathies. Our cases suggest that this thrombotic adverse event should not be overlooked, and coagulation parameters need to be monitored during the course of this treatment.
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http://dx.doi.org/10.1111/1346-8138.14798DOI Listing
April 2019

Approach for the Derivation of Melanocytes from Induced Pluripotent Stem Cells.

J Invest Dermatol 2018 01 5;138(1):150-158. Epub 2017 Sep 5.

Institute of Dermatology & Cutaneous Sciences, Sapporo, Japan.

Induced pluripotent stem (iPS) cells have the ability to differentiate into multiple cell types in the body and have an unlimited growth potential. However, iPS cell-derived melanocytes produced by existing protocols have significant limitations in developing novel strategies for regenerative medicine and cell therapies of pigmentation disorders in humans because they involve culture in media containing fetal bovine serum and nonphysiological agents. In this study, we established an in vitro approach to generate iPS cell-derived human melanocytes that have higher proliferation rates and increased melanin production compared with melanocytes prepared by previously reported approaches. Importantly, our iPS cell-derived human melanocytes are prepared in fetal bovine serum-free culture conditions that do not contain any nonphysiological agents. We designed two original methods, transferring black colonies by pipette and recovering black cell pellets from centrifuged medium, and numerous human iPS cell-derived melanocytes proliferated in gelatinous dishes coated with Matrigel after 12 days. We also succeeded in inducing melanin pigmentation in the nude mouse skin in vivo using those human iPS cell-derived melanocytes. We propose that this method using iPS cells established from T cells in the blood of normal human volunteers could be applied clinically to develop regenerative medicine and cell therapies for various forms of human pigmentation disorders.
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http://dx.doi.org/10.1016/j.jid.2017.07.849DOI Listing
January 2018

Elevated levels of serum IgM anti-phosphatidylserine-prothrombin complex antibodies in patients with cancer-associated vasculitis.

Int J Dermatol 2017 10 19;56(10):e203-e204. Epub 2017 Jun 19.

Department of Dermatology, St. Marianna University School of Medicine, Kawasaki, Japan.

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http://dx.doi.org/10.1111/ijd.13689DOI Listing
October 2017

Elevated moesin mRNA level in skin tissue of patients with polyarteritis nodosa based on real time RT-PCR.

J Dermatol Sci 2017 Jul 15;87(1):94-97. Epub 2017 Feb 15.

Department of Health Protection, Graduate School of Medicine, Teikyo University Asia International Institute of Infectious Disease Control, Japan.

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http://dx.doi.org/10.1016/j.jdermsci.2017.02.002DOI Listing
July 2017

Importance of appropriate location and frequency of biopsy for cutaneous manifestations in eosinophilic granulomatosis with polyangiitis.

Int J Dermatol 2016 Dec 8;55(12):1388-1390. Epub 2016 Jan 8.

Department of Dermatology, St. Marianna University School of Medicine, Kawasaki, Japan.

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http://dx.doi.org/10.1111/ijd.13210DOI Listing
December 2016

Presence of anti-phosphatidylserine-prothrombin complex antibodies and anti-moesin antibodies in patients with polyarteritis nodosa.

J Dermatol 2017 Jan 27;44(1):18-22. Epub 2016 Jun 27.

Department of Dermatology, St Marianna University School of Medicine, Kawasaki, Japan.

We measured both serum anti-phosphatidylserine-prothrombin complex (anti-PSPT) antibodies and anti-moesin antibodies, as well as various cytokines (interleukin [IL]-2, IL-4, IL-5, IL-10, IL-13, IL-17, granulocyte macrophage colony-stimulating factor, γ-interferon, tumor necrosis factor-α) levels in polyarteritis nodosa (PAN) patients with cutaneous manifestations. All patients showed the presence of a histological necrotizing vasculitis in the skin specimen. They were treated with i.v. cyclophosphamide pulse therapy (IV-CY) and prednisolone therapy or steroid pulse therapy. The immunological assessments were performed on sera collected prior to and after treatment with IV-CY or steroid pulse therapy. We found a significant positive correlation between serum anti-moesin antibodies and both clinical Birmingham Vasculitis Activity Scores and Vasculitis Damage Index. Anti-PSPT antibody and IL-2 levels after treatment in PAN patients were significantly lower than before treatment. In contrast, anti-moesin antibody levels were higher following IV-CY or steroid pulse therapy compared with the pretreatment levels. In the treatment-resistant PAN patients (n = 8), anti-PSPT antibody levels after treatment were significantly lower than before treatment. In contrast, anti-moesin antibody levels after treatment in the patients were significantly higher compared with the pretreatment levels. Immunohistochemical staining revealed moesin overexpression in mainly fibrinoid necrosis of the affected arteries in the PAN patients. We suggest that measurement of serum anti-PSPT antibody levels could serve as a marker for PAN and aid in earlier diagnosis of PAN. We also propose that elevated serum anti-moesin antibodies could play some role of the exacerbation in patients with PAN.
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http://dx.doi.org/10.1111/1346-8138.13491DOI Listing
January 2017

Therapeutic effect of autologous platelet-rich plasma (PRP) on recalcitrant cutaneous ulcers in livedoid vasculopathy.

JAAD Case Rep 2015 Sep 25;1(5):310-1. Epub 2015 Aug 25.

Department of Dermatology, St. Marianna University School of Medicine, Kawasaki, Japan.

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http://dx.doi.org/10.1016/j.jdcr.2015.07.005DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC4809273PMC
September 2015

Complete resolution of refractory cutaneous arteritis by intravenous cyclophosphamide pulse therapy.

Int J Dermatol 2015 Aug 3;54(8):e323-5. Epub 2015 Jul 3.

Department of Dermatology, St. Marianna University School of Medicine, Kawasaki, Japan.

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http://dx.doi.org/10.1111/ijd.12863DOI Listing
August 2015

Proposal of quality indicators for cutaneous vasculitis.

J Dermatol 2014 Aug 2;41(8):755-6. Epub 2014 Jul 2.

Department of Dermatology, St Marianna University School of Medicine, Kawasaki, Japan.

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http://dx.doi.org/10.1111/1346-8138.12545DOI Listing
August 2014

Treatment for cutaneous arteritis patients with mononeuritis multiplex and elevated C-reactive protein.

J Dermatol 2013 Dec 4;40(12):955-61. Epub 2013 Dec 4.

Department of Dermatology, St Marianna University School of Medicine, Kawasaki, Japan.

Cutaneous arteritis (cutaneous polyarteritis nodosa, CA) is a necrotizing vasculitis of arteries within the skin. CA is a new classification under single-organ vasculitis, as adopted by the 2012 Chapel Hill consensus conference (CHCC 2012). Some patients originally diagnosed as having CA could develop additional disease manifestations that warrant reclassifying as systemic polyarteritis nodosa (PAN) according to the CHCC 2012. We retrospectively investigated 101 patients with CA seen at our department between 2003 and 2012. There was a significantly higher frequency of inflammatory plaques and leg edema in CA patients with elevated C-reactive protein (CRP) compared to CA patients with normal CRP. Similarly, there were significant differences in the incidence of arthralgia and mononeuritis multiplex between the two patient groups. We found significantly positive correlations between CRP and creatinine titers in serum in all 101 CA patients. Prednisolone was administrated in a significantly greater percentage of patients with elevated CRP compared to patients with normal CRP. Repeated i.v. cyclophosphamide pulse therapy (IV-CY) with prednisolone therapy at an early stage resulted in complete resolution without adverse effects or severe complications. We regard inflammatory plaques and leg edema with elevated serum CRP as an indication of a more severe condition, and treated them effectively with prednisolone. Assuming mononeuritis multiplex and/or arthritis exist with elevated CRP, we propose that earlier treatment by IV-CY with prednisolone should be indicated for CA patients who demonstrate these more severe manifestations to prevent progression to PAN.
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http://dx.doi.org/10.1111/1346-8138.12303DOI Listing
December 2013

Significance of two skin biopsy performances with consecutive deeper sections in the differential diagnosis between cutaneous polyarteritis nodosa and livedo vasculopathy.

Acta Derm Venereol 2014 Jan;94(1):84-5

Department of Dermatology, St. Marianna University School of Medicine, 2-16-1 Sugao, Miyamae-ku, Kawasaki, Kanagawa 216-8511, Japan.

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http://dx.doi.org/10.2340/00015555-1603DOI Listing
January 2014

Lysosomal-associated membrane protein-2 plays an important role in the pathogenesis of primary cutaneous vasculitis.

Rheumatology (Oxford) 2013 Sep 23;52(9):1592-8. Epub 2013 May 23.

Department of Dermatology, St Marianna University School of Medicine, Miyamae-ku, Kawasaki, Kanagawa, Japan.

Objectives: Recent research suggests that lysosomal-associated membrane protein-2 (LAMP-2) could be one of the target antigens in the pathogenesis of vasculitides. We established a transgenic rat model, env-pX rats, with various vasculitides including cutaneous vasculitis. Human primary cutaneous vasculitis includes cutaneous polyarteritis nodosa (CPN) and Henoch-Schönlein purpura (HSP). We measured serum anti-LAMP-2 antibody levels in morbid env-pX rats and injected anti-LAMP-2 antibody into premorbid env-pX rats. We further measured serum anti-LAMP-2 antibody levels in patients with CPN and HSP.

Methods: Cutaneous vasculitis was observed in ∼30% of 6-month-old morbid env-pX rats. In contrast, these findings were rare in premorbid env-pX rats under 3 months old. We also examined 85 patients with CPN and 36 adult patients with HSP. Serum anti-LAMP-2 antibody levels were determined using ELISA. Premorbid env-pX rats under 3 months old were given an i.v. injection of anti-LAMP-2 antibody at day 0 and day 7. At day 14, these rats underwent histopathological and direct immunofluorescence examination. Cell surface LAMP-2 expression of rat neutrophils was examined by flow cytometry.

Results: Serum anti-LAMP-2 antibody levels were significantly higher in morbid env-pX rats than in wild-type normal rats. In addition, the levels in the cutaneous vasculitis group of morbid env-pX rats were significantly higher than the no cutaneous vasculitis group. Intravenous anti-LAMP-2 antibody injection into premorbid env-pX rats under 3 months old induced infiltration of neutrophils into cutaneous small vessels. Anti-LAMP-2 antibody-binding neutrophils were detected there. LAMP-2 expression on the cell surface of neutrophils in premorbid env-pX rats under PMA stimulation was higher compared with controls. Serum anti-LAMP-2 antibody levels in CPN and HSP were significantly higher than those of healthy controls.

Conclusion: These data support a positive relationship between anti-LAMP-2 antibody and cutaneous vasculitis.
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http://dx.doi.org/10.1093/rheumatology/ket159DOI Listing
September 2013

Relationship among antineutrophil cytoplasmic antibody, blood urea nitrogen and complement in patients with eosinophilic granulomatosis polyangiitis (Churg-Strauss syndrome).

J Dermatol 2013 Jul 17;40(7):511-5. Epub 2013 Apr 17.

Department of Dermatology, St Marianna University School of Medicine, Kawasaki, Japan.

Eosinophilic granulomatosis with polyangiitis (EGPA), also known as Churg-Strauss syndrome, is an antineutrophil cytoplasmic antibody (ANCA)-associated vasculitis characterized by a history of asthma, hypereosinophilia. The prevalence of ANCA in EGPA is less common than in other ANCA-associated vasculitis. Increasing evidence of complement activation in the pathogenesis of ANCA-associated vasculitis has been provided by studies in animal models. We examined EGPA patients with cutaneous manifestations as an initial sign and investigated the correlations among clinical, serological and histopathological findings. We focused on differences among ANCA, blood urea nitrogen and complement levels such as complement 3 (C3), C4 and total complement hemolytic activity (CH50). We retrospectively investigated the records of 22 patients (11 male and 11 female) with EGPA admitted to our hospital from 1997-2012. Ten of the 22 patients (46%) were positive for serum myeloperoxidase (MPO)-ANCA. In contrast, all the patients were negative for serum proteinase 3 ANCA. There was a significantly positive correlation between serum CH50 and C4 levels in patients with EGPA. Serum blood urea nitrogen (BUN) levels differed significantly between MPO-ANCA-positive and -negative patients. Serum CH50 levels were higher in MPO-ANCA-positive patients compared to negative patients. Serum BUN levels were higher in elevated CH50 patients compared to normal and low CH50-negative patients. We propose that positive findings for MPO-ANCA with CH50 high activity may be a risk factor for developing renal insufficiency. Assuming there are correlations between the presence of ANCA and complements, earlier diagnosis based on initial efficacious treatment for EGPA.
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http://dx.doi.org/10.1111/1346-8138.12163DOI Listing
July 2013

IgM in lesional skin of adults with Henoch-Schönlein purpura is an indication of renal involvement.

J Am Acad Dermatol 2010 Dec 8;63(6):1026-9. Epub 2010 Oct 8.

Department of Dermatology, St Marianna University School of Medicine, Kawasaki, Kanagawa, Japan.

Background: Henoch-Schönlein purpura (HSP) is a multisystem disease believed to be a consequence of the entrapment of circulating IgA-containing immune complexes in blood vessel walls throughout the skin, kidneys, and gastrointestinal tract. The skin manifestations are characterized by nonthrombocytopenic palpable purpura over the lower extremities.

Objective: We assessed adult patients with HSP who had nonthrombocytopenic palpable purpura on the extensor surfaces of their lower limbs, and had no associated connective tissue disease. Patient medical records, including clinical presentation, laboratory data, and direct immunofluorescence (DIF) reports, were reviewed retrospectively.

Methods: We reviewed the records of 25 adult patients with HSP who presented at our department, between 2006 and 2008, with an initial cutaneous manifestation of palpable purpura on their lower extremities. Adult HSP was defined in all cases as documented leukocytoclastic vasculitis according to a skin biopsy specimen, with histopathologic evidence of IgA deposition by DIF. Statistical analyses were performed using a χ(2) test to compare prevalence among each clinical manifestation.

Results: There was a significant correlation between IgM deposition by DIF and renal involvement (χ(2) = 5.23, P = .022). IgM deposition and complement 3 deposition by DIF showed a close relationship (χ(2) = 5.11, P = .024). There was a significant positive correlation between serum IgA and C-reactive protein levels (Spearman's rank correlation coefficient = 0.35, P = .044).

Limitations: These findings should be validated in larger studies. Renal biopsies were not done to confirm the presence of nephritis.

Conclusions: This study suggests that IgM deposition in palpable purpura based on DIF provides an indicator of nephritis in adult patients with HSP. We believe that IgM deposition could be related to the pathogenic factors that trigger the development of renal involvement.
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http://dx.doi.org/10.1016/j.jaad.2009.11.690DOI Listing
December 2010

Drug-induced hypersensitivity syndrome: drug reaction with eosinophilia and systemic symptoms (DRESS) syndrome induced by aspirin treatment of Kawasaki disease.

J Am Acad Dermatol 2009 Jan;60(1):146-9

Department of Dermatology, St. Marianna University School of Medicine, Kawasaki, Kanagawa, Japan.

Drug-induced hypersensitivity syndrome (DIHS), also known as drug reaction with eosinophilia and systemic symptoms (DRESS), is a severe multiple-organ condition caused by drug treatment. The current report describes a Japanese boy who underwent aspirin treatment for Kawasaki disease, and who subsequently presented with the manifestations of DIHS/DRESS syndrome. He had been treated with a single high dose of intravenous immunoglobulin and aspirin orally for Kawasaki disease. One month after the onset of Kawasaki disease, he developed a generalized maculopapular eruption, high-grade fever, leukocytosis with eosinophilia, and an increased number of atypical lymphocytes, severe liver dysfunction, lymphadenopathy, and prominent increases in antihuman herpesvirus-6 immunoglobulin G titer. The activity of 2',5'-oligoadenylate synthetase was elevated at the onset stage. Hypersensitivity to aspirin was confirmed by skin patch test and by lymphocyte stimulation test. Based on these findings, the patient was diagnosed with DIHS/DRESS caused by aspirin. To our knowledge, there have been no previous reports of aspirin-induced hypersensitivity syndrome subsequent to Kawasaki disease. The activity of 2',5'-oligoadenylate synthetase might be useful as a diagnostic marker of DIHS/DRESS syndrome and for exploring its pathogenesis.
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http://dx.doi.org/10.1016/j.jaad.2008.07.044DOI Listing
January 2009
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