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ABSTRACT: Lupus cystitis is a rare complication associated with systemic lupus erythematosus (SLE). This rare disorder has been reported mainly from East Asia, especially from Japan. In this study, we describe a patient with lupus cystitis and review 37 reported cases in Japan. Bowel symptoms preceded urinary symptoms in 13 of 37 (35%) patients, whereas five patients complained of only bowel symptoms. Hydronephrosis was detected in 34 of 37 patients (92%). The possibility of lupus cystitis should be considered when patients complain of bowel or urinary symptoms. Examination for hydronephrosis is critical for diagnosis.
Lupus 02/2009; 18(7):655-8. · 2.34 Impact Factor
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ABSTRACT: We report a 29-year-old woman with a 15-year history of recurrent pruritic urticarial erythemas. The individual lesions lasted for > 24 h, and antihistaminic agents were not effective. Histological examination of a skin biopsy revealed interstitial oedema of the dermis and perivascular infiltration of numerous eosinophils without vasculitis. No internal organ involvement or peripheral blood eosinophilia was present. A diagnosis of persistent urticaria was made and the patient was successfully treated with oral corticosteroid therapy. Persistent urticaria has been described as an unusual reaction that lasts longer than typical urticaria. It is effectively treated with corticosteroids, but not with antihistaminic agents. In order to choose the most effective treatment, persistent urticaria should be recognized as a different clinical condition from typical urticaria.
Clinical and Experimental Dermatology 12/2008; 34(5):e14-7. · 1.20 Impact Factor
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ABSTRACT: As Bowen's disease of the nail apparatus is quite rare, there have been only a few reports on the prevalence of human papillomavirus (HPV) infection in this condition.
The purpose of this study was to clarify the association of HPV with this disease involving the nail apparatus.
Five patients with Bowen's disease of the nail apparatus were investigated clinically, virologically and histologically. Total DNAs extracted from excised skin lesions were analysed using polymerase chain reaction (PCR) for the presence of HPV DNA and the amplified products were subjected to DNA sequence analyses. Histological localization of HPV DNA was examined by in situ hybridization.
In three of five patients, HPV was detected by PCR amplification, and subsequent sequence analyses of the PCR products showed the sequences of HPV type 56. A common clinical feature of the three HPV-positive patients was longitudinal melanonychia. In contrast, the two HPV-negative patients presented with a convex nail deformity and a periungual ulcerative lesion. In two of three positive cases, there was a silent point mutation in the L1 gene of each HPV. In the remaining one case, the nucleotide sequence was consistent with the consensus sequence of HPV 56. Sequence analyses of the E6 gene revealed the infection of different variants of HPV 56 among the three cases. The viral genomes were located in keratinocyte nuclei upon in situ hybridization.
HPV 56 may be involved in the carcinogenesis of Bowen's disease affecting the nail matrix with longitudinal pigmentation.
British Journal of Dermatology 07/2008; 158(6):1273-9. · 3.67 Impact Factor
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ABSTRACT: Animal models are useful tools to study various aspects of human diseases. Bleomycin (BLM)-induced scleroderma mouse has been widely investigated as an animal model of scleroderma. Repeated injections of BLM, either daily or every other day, for 3-4 weeks are required to induce scleroderma in mice. Poly(L-lactic acid) (PLA) is a biodegradable, biocompatible and bioabsorbable device that has been widely investigated for controlled drug release. In this study, we fabricated BLM-containing PLA microspheres and subcutaneously injected them into C3H mice for only one time.
Treated skins were harvested at days 7 and 21. Then, histological examination and collagen content measurement assay were performed. The mRNA expression of alpha1(I) collagen (COL1A1), monocyte chemoattractant protein-1 (MCP-1), TGF-beta(1) and connective tissue growth factor (CTGF) were quantified by real-time PCR.
Dermal fibrosis was histologically observed at day 7 after injection and remained present at day 21. Tissue responses against BLM-PLA microspheres alone were mild. Soluble collagen content and expression level of alpha1(I) collagen mRNA were significantly elevated at day 21. Expression levels of MCP-1 mRNA and TGF-beta(1) mRNA at day 7 and CTGF mRNA at day 21 were also elevated.
The present study demonstrated for the first time that one-time injection of BLM-PLA microspheres can induce dermal fibrosis in C3H mice. BLM-PLA microspheres thus offer a labour-saving, simple and powerful tool to establish an animal model of BLM-induced dermal fibrosis.
Rheumatology (Oxford, England) 05/2008; 47(4):454-7. · 4.24 Impact Factor
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Journal of the European Academy of Dermatology and Venereology 04/2008; 22(11):1392-4. · 2.98 Impact Factor
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ABSTRACT: Although digital ulcerations frequently occur in patients with systemic sclerosis (SSc), there have been few reports on the macrovascular involvement.
To evaluate the macrovascular involvement in patients with SSc exhibiting digital ulceration or gangrene.
Transfemoral catheter arteriography of the upper and/or lower extremities was performed in eight patients (one man and seven women, age range 42-71 years) with SSc exhibiting digital ulceration or gangrene. The background of the patients, such as autoantibody profiles and vascular risk factors including smoking habits, was also investigated.
Macrovascular involvement was detected in seven of eight patients. In three of seven patients who underwent arteriography of the upper extremity, occlusion was limited to the digital arteries. Obliteration of the ulnar artery and superficial palmar arch was detected in three of seven patients, and the radial artery in one patient. Only one of five patients who underwent arteriography of the lower extremity showed the occlusion limited to digital arteries of the foot. Occlusion of the posterior tibial artery, dorsalis pedis artery and arcuate artery was detected, each in one patient. Two patients showed occlusion of the plantar arch. Overall, the occlusion of arteries proximal to the digits was demonstrated in four of eight patients. Three of the four patients were positive for antitopoisomerase-1 antibody and had diffuse cutaneous SSc (dcSSc) with multiple skin ulcers or gangrene.
Macrovascular involvement as detected with arteriography is not rare in SSc patients with digital ulceration or gangrene. Moreover, the vascular occlusion proximal to the digits seemed to be frequent in antitopoisomerase-1 antibody-positive dcSSc patients with multiple skin ulcers or gangrene.
British Journal of Dermatology 01/2007; 155(6):1159-64. · 3.67 Impact Factor
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ABSTRACT: Human skin shows various morphological characteristics, depending on the body site. As these distinct phenotypes have been explained on the basis of the variance in epidermal keratinocytes and the presence of skin appendages, the spatial distinction of the dermal components has not been fully elucidated.
To identify and characterize the profiles of mRNAs that are abundantly or specifically expressed by fibroblasts derived from trunk skin, but not from palmoplantar skin or oral mucosa.
In order to identify the distinct mRNA expression by trunk skin fibroblasts, a subtraction cDNA screening was performed first, followed by Northern blotting, Western blotting and immunohistochemistry for cultured human and rat dermal fibroblasts and those skin tissues. Finally, whole mount in situ hybridization (WISH) was performed to examine the differences in the expression of the corresponding gene during the developmental stage of mouse embryos.
We identified three cDNA clones encoding fibronectin (FN), pregnancy-specific beta1-glycoprotein 5 and beta-actin, respectively, whose mRNAs were abundantly or specifically expressed by trunk skin fibroblasts. FN and some integrins were further confirmed to be expressed more selectively in human and rat trunk skin fibroblasts, both in terms of the RNA and the protein levels, compared with the fibroblasts derived from plamoplantar skin and oral mucosa. WISH demonstrated that FN was localized around the hair follicles of mouse embryos.
FN, one of most potent extracellular matrix molecules, was demonstrated to be spatially transcribed depending on the body sites. The distinct expression of FN was suggestive of the essential commitment in the process of cutaneous development and morphogenesis of appendages originated from hair germ. The paucity of FN in palmoplantar skin and oral mucosa might explain the characteristics of these skin phenotypes.
British Journal of Dermatology 10/2006; 155(3):522-31. · 3.67 Impact Factor
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ABSTRACT: We describe two cases of a 3-year-old Japanese boy and his 1-year-old sister presenting recessive dystrophic epidermolysis bullosa; a relatively mild phenotype. Blistering and scarring were limited to the acral region, and some fingernails and toenails were lost. PCR-RFLP and DNA sequencing analyses revealed compound heterozygotes for a splice-site mutation (6573 +1GtoC) and a nonsense mutation (E2857X) in the type VII collagen gene (COL7A1). Both mutations caused a premature termination codon (PTC). The mutation E2857X was located behind the candidate cleavage site within the NC-2 domain required for the assembly of anchoring fibrils. This PTC position may explain their mild phenotype.
International Journal of Dermatology 04/2006; 45(3):302-5. · 1.14 Impact Factor
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ABSTRACT: We report a 27-year-old Japanese man with the peculiar clinical features of acrogeria. He had had perniosis since early childhood. Prominent atrophic skin changes over the hands, hallux valgus, shortened distal phalanges and atrophic scars on his auricles were noted. X-ray of the hands revealed acro-osteolytic changes of the distal phalanges, and arteriography demonstrated multiple occluded branches of the digital arteries. There were no histological changes of systemic sclerosis in his forearm skin, nor antinuclear antibodies or coagulation disorders. Western immunoblotting demonstrated decreased production of type III collagen by dermal fibroblasts both from an affected finger and from the unaffected upper arm. Although the pathogenesis of acrogeria is unknown, the present case suggests that peripheral circulatory disturbance, as well as a congenital abnormality in type III collagen synthesis, may partly account for the pathogenesis of Gottron-type acrogeria.
British Journal of Dermatology 09/2004; 151(2):497-501. · 3.67 Impact Factor
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ABSTRACT: We describe the case of a 59-year-old Japanese woman presenting with generalized lentigines without systemic anomalies. She had a medical history of gastrointestinal stromal tumors (GISTs), in which gain-of-function mutations of the c-kit gene had recently been found. We detected a point mutation at codon 557 in exon 11 of leukocyte DNA from the patient. The stem cell factor-type III receptor tyrosine kinase pathway plays important roles in the regulation of melanocyte proliferation and differentiation. We speculate that the generalized lentigines of the patient may be caused by melanocyte proliferation due to the c-kit gene mutation.
Dermatology 02/2004; 208(3):217-20. · 2.05 Impact Factor
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ABSTRACT: Microchimerism of foetal cells occurs during most pregnancies. Two autoimmune diseases, systemic sclerosis (SSc) and Sjögren's syndrome (SS), have many clinical and pathological similarities to chronic graft-vs-host disease (GVHD). These findings suggest that anti-maternal graft-vs-host reaction by foetal cells may be involved in the pathogenesis of the diseases. To explore this hypothesis, we examined foetal DNA in peripheral blood of 59 women and in salivary glands from 28 women.
DNA extracted from peripheral blood and the affected minor salivary glands was analysed for the Y-chromosome-specific gene using a nested polymerase chain reaction (PCR) test. In the minor salivary gland specimens, the Y-chromosome-positive foetal cells were identified by in situ hybridization with a Y-chromosome-specific DNA probe.
In peripheral blood, there was no significant difference between controls and patients with SSc or SS. In salivary glands, foetal DNA was detected in 11 of 20 women with SS but in only one of eight normal controls using PCR test. Additionally, foetal cells were clearly detected in three out of eight women with SS by the use of in situ hybridization.
The identification of foetal cells in salivary glands suggests that anti-maternal GVHD may be involved in the development of SS.
Rheumatology 06/2002; 41(5):490-5. · 4.06 Impact Factor
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ABSTRACT: To evaluate the effects of newly developed nitroglycerine (NTG) tape on the peripheral circulatory disturbance in systemic sclerosis (SSc).
The finger temperature (FT) of 25 patients with SSc was monitored by thermography before the application of NTG tape to the wrist and one hour afterwards. To check the placebo effect, the change of FT with placebo tape in six patients who had shown a distinct rise in FT (>2 degrees C) with NTG tape was monitored.
Twenty one patients with SSc had a low FT before the application of NTG tape (colder than mean-2SD of normal controls). FT was significantly raised in patients with SSc after the application of NTG tape (p<0.01). Patients with low FT showed a distinct rise in FT more often than patients with a normal FT (71% v 0%, p<0.01). FT was raised with NTG tape more than with placebo tape (4.2 (SD 1.9) degrees C v 2.2 (2.8) degrees C, p<0.05).
The application of NTG tape increases peripheral circulation in patients with SSc who have distinct circulatory disturbance. The application of NTG tape may improve peripheral circulatory disturbance in SSc.
Annals of the Rheumatic Diseases 02/2002; 61(2):177-9. · 8.73 Impact Factor
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ABSTRACT: We describe a 68-year-old healthy woman with an unusual skin manifestation of mycetoma. A dark reddish erythematous plaque was present on the dorsum of the left hand for over 10 years. A skin biopsy revealed sulphur granules in the abscess, and Nocardia nova was identified from the specimen. Treatment with dapsone resulted in significant improvement. We review the clinical features of cutaneous N. nova infections.
British Journal of Dermatology 08/2001; 145(1):154-6. · 3.67 Impact Factor
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European journal of dermatology: EJD 01/2001; 10(8):633-4. · 2.53 Impact Factor
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ABSTRACT: Histidinohydroxylysinonorleucine (HHL) is a trivalent cross-link of type I collagen that is stable and has physiologic properties. In this study, we conducted a biochemical quantification of HHL in five patients with morphea. We compared the quantity of HHL contained in the lesional skin and the adjacent normal skin. The molar ratio of HHL to collagen in the sclerotic lesion was significantly higher than that of the adjacent normal skin (0.36 +/- 0.09 vs. 0.28 +/- 0.10, P<0.01). We could find no relationship between an increase of HHL content and the duration of the disease. The results indicate that an increased amount of HHL may play a certain role to inducing the sclerotic skin change characteristic of morphea.
Journal of Dermatological Science 01/2001; 25(1):83-6. · 3.72 Impact Factor
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ABSTRACT: We report a 53-year-old man with a 2-year history of a violaceous indurated plaque on the shoulder. Although angiosarcoma was clinically suspected, histological examination revealed numerous lobules ('tufts') with cleft-like vascular lumina throughout the dermis and subcutaneous tissue. Tumour cells had no nuclear atypia and were positive for CD34, but almost negative for factor VIII-related antigen. These findings were compatible with a diagnosis of tufted angioma, or angioblastoma. We reviewed 41 cases reported in Japan and found that, although most patients presented during the first year of life (23/41), the condition does occur throughout childhood and adult life. Both sexes are affected equally and, contrary to some reports, it is unlikely that oestrogens have a pathogenic role.
Clinical and Experimental Dermatology 12/2000; 25(8):627-30. · 1.20 Impact Factor
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ABSTRACT: The effect of hypoxia on the expression of extracellular matrix-related genes by human dermal fibroblasts was investigated using a novel three-dimensional culture supplemented with L-ascorbic acid 2-phosphate. Experiments were performed by placing replicate dishes in either hypoxic (2%) or in normoxic (20%) condition for various periods of time ranging up to 72 h. The mRNA expression levels of proalpha1(I), proalpha1(III) collagens and MMP-1 were analyzed using Northern blotting. Hypoxia transiently increased proalpha1(I) and proalpha1(III) collagen gene expression at 24 h, but a prolonged exposure to hypoxia decreased them. A slight increase in MMP-1 mRNA was observed at 24 h and prolonged exposure for up 72 h resulted in significantly increased expression of MMP-1 gene. Our results suggest that enhanced degradation as well as decreased synthesis of collagens induced by hypoxia may account for the delayed wound healing associated with circulatory disturbances.
Journal of Dermatological Science 12/2000; 24(2):99-104. · 3.72 Impact Factor
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ABSTRACT: To know whether cultured human mast cells raised from umbilical cord blood cells in the presence of stem cell factor (SCF) and interleukin-6 (IL-6) can be a model of human skin mast cells, the cells were stimulated, and intracellular calcium ion ([Ca(2+)](i)) mobilization was analyzed by fluorescence microscopic techniques in parallel with a measurement of histamine released from the cells. When IgE-sensitized mast cells were activated by anti-IgE, [Ca(2+)](i) elevation began at the periphery and subsequently proceeded toward the center of the cells. The increase in [Ca(2+)](i) in calcium ionophore A23187-stimulated mast cells began at the center and spread to the periphery of the cells. Significant histamine release was observed by each stimulation. However, either compound 48/80 or substance P failed to increase [Ca(2+)](i) with no appreciable histamine release. This study shows that there is heterogeneity of [Ca(2+)](i) mobilization in the activated human mast cells, and that cultured human mast cells derived from umbilical cord blood cells in the presence of SCF and IL-6 can not be a model of human skin mast cells.
Journal of Dermatological Science 12/2000; 24(2):146-52. · 3.72 Impact Factor
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ABSTRACT: Human mast cells are classified into two phenotypes based on their neutral protease compositions. One type is a tryptase-positive and chymase-positive MCTC cell that is predominant in the skin, another is a tryptase-positive and chymase-negative MCT cell that is predominant in the lung. Cord blood-derived human mast cells cultured in the presence of stem cell factor and interleukin-6 are a mixture of MCTC and MCT at various ratios, as revealed by immunocytochemical staining. We performed an electron microscopic analysis of cord blood-derived human cultured mast cells and found that they were so immature that we could not distinguish MCT and MCTC from their ultrastructural morphology. The response to secretagogues was not the response of MCTC but rather of MCT. Although human cultured mast cells are the most useful cells for use in in vitro experiments, the present culture condition supplemented with stem cell factor and interleukin-6 does not develop fully mature mast cells in vitro.
Journal of Dermatological Science 10/2000; 24(1):4-13. · 3.72 Impact Factor
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ABSTRACT: Possible involvement of mast cells in pulmonary sarcoidosis has been suggested, however whether mast cells are involved in cutaneous sarcoidosis remains unknown. We undertook a morphological study of mast cells in the lesional skin from 17 patients with cutaneous sarcoidosis using immunohistochemical methods. Mast cells were present in non-parenchymal fibrous areas, but not in granulomatous areas, in the biopsy specimens from the cutaneous lesions. However, there were no significant differences between the number of mast cells in the lesional skin and that in non-lesional skin from the patients. Mast cells containing substantial quantities of both tryptase and chymase (MC(TC) cells) were present in 41% of the patients, and cells containing tryptase but not chymase (MC(T) cells) were present in 59% of patients. All patients of the former group showed systemic manifestations of the disease concomitantly. Serum angiotensin I-converting enzyme levels were elevated in 71.4% of the former group, and in 30% of the latter group. This study for the first time demonstrated that mast cells were present in non-parenchymal fibrous areas of the cutaneous lesions of sarcoidosis, and the mast cell subtypes may be related to systemic manifestations.
Journal of Dermatological Science 10/2000; 24(1):60-6. · 3.72 Impact Factor
