Search PubMed⌕ Search

Biomedical subjects

Y Horiguchi

Publications and source records attributed to Y Horiguchi.

At least 181 records · Page 10Linked to original sources

Epidermolysis bullosa simplex: identification of a kindred with autosomal recessive transmission of the Weber-Cockayne variety.

With few exceptions, epidermolysis bullosa simplex (EBS) is transmitted as an autosomal dominant trait. All cases of autosomal recessive EBS reported to date have been associated with significant extracutaneous disease (including anemia, marked growth retardation, dentition abnormalities, and/or concurrent neuromuscular disease), early infant mortality, or often, rather extensive cutaneous involvement. In some, the cutaneous morphology has even suggested junctional or dystrophic disease. We cared for a kindred in which four individuals were affected with an autosomal recessive form of EBS with cutaneous findings most often suggestive of Weber-Cockayne disease. Except for scattered oral erosions in one patient, there was no evidence of associated extracutaneous disease. These findings demonstrate the increasing complexity of even the simplex forms of inherited epidermolysis bullosa. This obviously has implications for genetic counseling.

Adolescent↗

Kindler syndrome: report of two cases and review of the literature.

We evaluated two patients with hereditary bullous poikiloderma. Both had acral bullae, generalized poikiloderma with prominent atrophy, and acral keratoses. One patient, with sporadic disease, had, in addition, urethral and subglottic stenoses, webbing of digits, and poor dentition. The other patient, whose disease was dominantly inherited, had koilonychia. The results of cutaneous histopathology, electron microscopy, and immunofluorescence mapping studies are presented. It is possible that Kindler syndrome and Weary's hereditary acrokeratotic poikiloderma are variants of the same disease.

Age Factors↗

Responsiveness of the ductus arteriosus to prostaglandin E1 assessed by combined cross sectional and pulsed Doppler echocardiography.

Cross sectional echocardiography combined with Doppler echocardiography was used to record either ductal morphology or the flow profile within the ductus arteriosus before and after infusion of prostaglandin E1 in 25 newborn infants with cyanotic and acyanotic congenital heart disease with ductus dependent blood flow. The ultrasound results were compared with changes in arterial oxygen tension and the overall clinical response to prostaglandin E1 seen during the same period in 24 of the 25 patients in whom the degree of ductal narrowing could be determined with the ultrasound method. At the time of the study, the ductus was widely patent or slightly narrowed in 12 patients and was closed in two patients. These patients did not respond to prostaglandin E1. There was prominent localised narrowing of the ductus in seven patients and generalised narrowing in three. After the infusion of prostaglandin E1 there was no ductal narrowing in these patients, except for one patient who had slight residual localised narrowing. There was also a considerable change in the ductal flow profiles in each patient. In these 10 patients infusion of prostaglandin E1 resulted in an increase in arterial oxygen tension, clinical improvement, or both. The present study indicates that prostaglandin E1 is effective in patients with prominent narrowing of the ductus but is not in patients in whom the ductus is widely patent or closed. Cross sectional echocardiography combined with Doppler echocardiography was useful for predicting the responsiveness of the ductus arteriosus to the infusion.

Alprostadil↗

Distribution, ultrastructural localization, and ontogeny of the core protein of a heparan sulfate proteoglycan in human skin and other basement membranes.

A variety of heparan sulfate proteoglycans (HSPG) have been identified on cell surfaces and in basement membrane (BM). To more fully characterize HSPG in human skin BM, we used two monoclonal antibodies (MAb) directed against epitopes of the core protein of a high molecular weight HSPG isolated from murine EHS tumor. Indirect immunofluorescence revealed linear distribution of HSPG within all skin BM, and within BM of all other human organs investigated. In a study of the ontogeny of HSPG in human skin BM, HSPG was detectable as early as 54 gestational days, comparable with other ubiquitous BM components, such as laminin and type IV collagen. Immunoelectron microscopy on adult skin and neonatal foreskin showed staining primarily within the lamina densa (LD) and sub-lamina densa regions of the dermoepidermal junction (DEJ) and vascular BM. In neonatal foreskin, additional staining was noted of basilar cytoplasmic membranes of keratinocytes, endothelial cells, and pericytes. We conclude that the core protein of a high molecular weight HSPG is ubiquitous in human BM, appears in fetal skin on or before 54 days, and is present primarily in the regions of the LD and sub-LD.

Antibodies, Monoclonal↗

[Breast carcinoma with features of osteosarcoma--a light and electron microscopic and immunohistochemical study].

Reported is the case history of a 58-year-old woman with a lump in the left breast. The tumor was composed of two nodules, sharing a common portion. Grossly, one was an irregular hard tumor that was grayish in color, and the other was a well circumscribed cystic tumor that showed necrosis and a hemorrhage that had filled it with a reddish soft mass. Light microscopically, combined features of a scirrhous carcinoma and of an osteosarcoma were observed in each nodule, but the common portion was consistent with a metaplastic carcinoma, with cancer cells and sarcoma-like cells closely mingled. An ultrastructural study showed that the sarcoma-like cells were composed of polymorphic cells that resembled osteoblasts, myofibroblasts, osteoclasts, histiocytes, and undifferentiated tumor cells. Immunohistochemically, vimentin and alpha 1-antitrypsin in the sarcoma-like cells were positive, suggesting these cells were of a mesenchymal rather than of an epithelial origin.

Adenocarcinoma, Scirrhous↗

[A case of mucinous carcinoma of the skin].

We were consulted by an 81-year-old man who had been complaining of a slow-growing tumor on his abdomen for 20 years. Histologically, small islets of the tumor cells were floating in mucinous lake separated by fibrous septa, so we diagnosed this tumor as mucinous carcinoma of the skin (Mendoza). Electron-microscopically, nuclei of the tumor cells were slightly folded. There were a few secretory granules and many secretory vacuoles in cytoplasm, but no findings of decapitation secretion. Like some other authors' reports, our data suggest that this tumor has an eccrine gland origin. To our knowledge, this is the 12th case of mucinous carcinoma of the skin in the Japanese literature.

Adenocarcinoma, Mucinous↗

[Clinical evaluation of synthetic hydroxyapatite implants for human periodontal osseous defects. 6 cases studied more than 3 years after the surgery].

We applied of granular hydroxyapatite HAP to periodontal bone defects, for the purpose of restoring the periodontal tissue destroyed by periodontal disease, the subjects were 6 patients (6 regions) with chronic marginal periodontitis who had passed more than 3 years since HAP implantation. After routine periodontal initial treatment, a flap operation was performed, and granular HAP, or a combination of granular and block-like HAP, was implanted into the periodontal bone defect. Clinical parameters were probing depth, clinical attachment level, clinical gingival margin, residual HAP volume and X-ray before operation, at 3, 6, 12 months, and 2, 3, and 4, 6 years after the operation. The results revealed that all measured values showed good results for both granular HAP and the combination of granular and block-like HAP after implantation, and that all cases showed stable results from 12 months on. No remarkable difference was noted between the implanted regions.

Bone Resorption↗

[An aged case of peripheral arterial obstructive disease with progressive limb necrosis].

An aged case of peripheral arterial obstructive disease with progressive limb necrosis is reported. A 70-year old man was admitted to our hospital with the complaint of numbness and rest pain on left lower limb. Thereafter, necrosis of the digital region of the upper limbs and the toe region of the lower limbs became progressively worse 3 weeks after onset. Bilateral lower limb amputations after lumbar sympathectomy and digital amputation of upper limbs after thoracic sympathectomy were performed. The histological findings of artery and vein in amputated limbs showed thromboangiitis obliterans. We conclude that Buerger's disease in the aged is rare, but does really exist.

Aged↗

[Successful CYVADIC combination chemotherapy of metastatic pulmonary lesions involving a mediastinal leiomyosarcoma--a case report].

Discussed is a 34-year-old man who hospitalized for manifesting the superior vena cava syndrome. Chest radiograph and chest CT scan revealed a gross tumor shadow in the superior mediastinum and a tiny shadow in the right lung. Superior vena cavography showed intimal stenosis, irregularities, and stiffness of the superior vena cava. Thus, radiotherapy applied to primary lesion of the mediastinum was provided to relieve his complaints. One year later, predominant metastatic pulmonary lesions and skin lesions developed. Light and electron microscopic findings of a biopsied specimen of the skin lesion were consistent with leiomyosarcoma. CYVADIC combination chemotherapy reduced the pulmonary metastatic lesions dramatically. The literature of mediastinal leiomyosarcoma is reviewed and the origin of this tumor is discussed.

Adult↗

Fulminant clonal expansion of large granular lymphocytes. Characterization of their morphology, phenotype, genotype, and function.

A 39-year-old woman exhibited abrupt malignant transformation of the large granular lymphocytes (LGL) after a chronic course of T gamma-lymphoproliferative disease (T gamma-LPD). The T gamma-lymphocytes were CD2+, CD3-, CD8-, CD16+, Leu7-, and Leu19+ with morphologic characteristics of LGL. Newly appearing LGL were much larger and had more prominent azurophilic granules. Although fundamentally they had the same phenotype as the LGL in chronic stage, they showed increased Ia-like antigen and decreased CD16 antigen expressions. Immunoglobulin (Ig) G-kappa type monoclonal component was detected in the patient's serum. The LGL showed a germ-line configuration for T-cell receptor (TCR) beta and gamma chain genes, whereas the clonal chromosomal abnormalities indicated the neoplastic nature of the LGL. The LGL exhibited competent natural killer (NK), interleukin 2 (IL2) activated killer (AK), and antibody-dependent cell-mediated cytotoxicity (ADCC) activities. The LGL may have derived from NK cells at their mature stage with prethymic phenotype and may have influenced the homeostasis of the patient's humoral immune response.

Adult↗

Cutaneous-type adult T-cell leukemia/lymphoma. A unique clinical feature with monoclonal T-cell proliferation detected by Southern blot analysis.

A 60-year-old woman suffered from multiple subcutaneous nodules with a self-limited clinical course. High titer of the antibody against the adult T-cell leukemia/lymphoma (ATL)-associated cell antigen was detected and atypical lymphocytes were present in less than 1% of the peripheral leukocytes. Tumor cells were identified by the molecular biology technique Southern blot analysis, which showed monoclonal cell expansion of the helper/inducer T cells integrated with human T-cell lymphotropic virus type I/adult T-cell leukemia virus. This patient was diagnosed as having the cutaneous type of smoldering ATL in a very early stage. In this case, only gene analysis of the skin lesion could facilitate making an early differential diagnosis of ATL from other lymphoproliferative diseases, including nonviral cutaneous T-cell lymphoma and benign lymphoid hyperplasia.

Antibodies, Monoclonal↗

A large inferior mesenteric-caval shunt via the internal iliac vein.

A large portosystemic shunt between the inferior mesenteric vein and the right internal iliac vein in a 28-yr-old non-cirrhotic man is presented. This collateral was discovered by ultrasound done as a screening examination for gastrointestinal bleeding. The direct communication of the inferior mesenteric vein with the internal iliac vein was demonstrated by computed tomography and percutaneous transhepatic portography. Surgical ligation of the collateral, performed to prevent future portosystemic encephalopathy, resulted in reduction of serum ammonia level and cessation of long-standing hemorrhoidal bleeding.

Adult↗

Letterer-Siwe disease: immunopathologic study with a new monoclonal antibody.

Three cases of Letterer-Siwe disease were studied with the monoclonal antibody Lag, which reacts to the antigen on the membranes of Birbeck granules and related structures of human Langerhans cells. Both lymph nodes and lesional skin contained abundant Lag-positive cells. By two-dimensional gel electrophoresis, antigenic substances in the lymph nodes of patients with Letterer-Siwe disease were found to have the same molecular weight of 40,000 dalton and isoelectric points extending from 4.7 to 6.5 as those in normal human skin and lymph nodes. Our results support the contention that Letterer-Siwe disease is a proliferative disorder of Langerhans cells. A double-staining method with Lag and anti-T6 antibody revealed that Lag reacted to 70% of T6-positive cells in the lymph nodes but to almost all such cells in skin lesions of patients with Letterer-Siwe disease, suggesting that the proliferating cells consist of at least Lag+, T6+, and Lag-, T6+ subpopulations.

Antibodies, Monoclonal↗

Characterization and distribution of prostaglandin D synthetase in rat skin.

The biochemical properties and immunohistochemical localization of prostaglandin D synthetase were investigated in adult rat skin. The activity of prostaglandin D synthetase, which isomerizes prostaglandin H2 to prostaglandin D2, was detected in the 100,000 g supernatant of the homogenate of adult rat skin. Whole skin showed considerable activity (1.9 nmol/min/mg protein), and prostaglandin D2 was the major prostaglandin among those formed from prostaglandin H2 in the presence of glutathione. The epidermis, which was separated from whole skin by heating (55 degrees C, 30 s), exhibited about three times higher activity (3.5) than the dermis (1.0). The enzymatic properties of both layers were similar; they were absolutely glutathione-dependent, were inhibited only a few percent by 1 mM 1-chloro-2,4-dinitro-benzene, and were completely absorbed by anti-rat spleen prostaglandin D synthetase antibody. Immunohistochemical studies, using anti-rat spleen prostaglandin D synthetase antibody and the immunoperoxidase method, showed that prostaglandin D synthetase was localized in Langerhans cells (not in keratinocytes) in the epidermis, in macrophages or histiocytes, and also in mast cells in the dermis. Immunoelectron microscopy also supported these findings. These results suggest that prostaglandin D2 is one of the most important arachidonic acid metabolites and plays a significant role in immunological function in the skin via Langerhans cells and macrophages.

Animals↗

Pathogenesis of lupus dermatoses in autoimmune mice. X. Evaluation of histamine-N-methyltransferase activity in the skin of autoimmune.

We measured histamine concentration and its metabolizing enzymes in the skin of MRL/Mp-lpr/lpr (MRL/l) and BXSB mice to clarify the contribution of histamine metabolism to the mechanisms of the development of lupus dermatoses. The concentration of histamine seemed to differ with the mouse strain. The activity of histamine-N-methyltransferase (HMT), one of two major metabolizing enzymes, was significantly lower in the tail and back skin of MRL/l mice at the age of 5 months than in the control MRL/Mp-+/+(MRL/n) mice, although there were no characteristic differences among several mouse strains of 1 mo of age. In the back skin of MRL/l mice, an age-dependent decrease of HMT activity was observed along with a corresponding decrease in histamine concentration, whereas an age-dependent increase of both HMT activity and histamine concentration was demonstrated in BXSB mice and other control mouse strains. Autoimmune-prone male BXSB mice and non-autoimmune female BXSB mice at 5 mo of age showed similar HMT activity. Corticosteroid treatment restored HMT activity in the skin of MRL/l mice but not in MRL/n mice. In addition, the change in HMT activity in MRL/l mice treated with corticosteroid appeared earlier than changes in clinicopathological examinations including skin eruptions, dermatopathology and proteinuria. Diamine oxidase (DAO) activity, another major metabolizing enzyme, was not detected in the skin of any autoimmune or control mouse strains. These findings suggest that the low activity of HMT in the skin of MRL/l mice plays a significant pathological role in the development of spontaneous lupus-like eruption. In other mouse strains, it is assumed that HMT activity is regulated by genetic factors.

Abdomen↗