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PubMed1996

Vitiligo in Vogt-Koyanagi-Harada disease: immunohistological analysis of inflammatory site.

BACKGROUND: Vogt-Koyanagi-Harada disease (VKH) consists of uveitis with associated non-ocular symptoms, such as vitiligo or central nervous system disorders. A cell-mediated immune disorder is believed to play an important role in VKH; however, the skin lesion has not been well studied. Therefore, for this study, we examined the skin of a 56-year-old Japanese man with Vogt-Koyanagi-Harada disease (VKH). METHODS: Skin biopsy specimens from the patient's vitiligo were obtained 1 month after the onset of ocular symptoms. Immunohistochemical analysis of the specimens study was performed using the following monoclonal antibodies: anti-T cell, anti-B cell, anti-major histocompatibility complex class II (HLA-DR), anti-T helper/inducer (CD4), and anti-T suppressor/ cytotoxic lymphocytes (CD8). RESULTS: Histopathologic analysis revealed mononuclear cell infiltration of the slightly edematous dermis, especially surrounding the hair follicles and sweat glands. Melaninladen cells in the epidermis were partially lost. The infiltrating mononuclear cells consisted primarily of T lymphocytes with a smaller number of B lymphocytes. Most showed expression of HLA-DR. CD4-positive lymphocytes were dominant over CD8-positive cells (3:1). CONCLUSIONS: The results indicate that vitiligo of VKH is infiltrated by mostly activated helper/inducer lymphocytes and that cell-mediated immunity plays an important role in the pathogenesis of the dermal lesions of VKH as well.

Antibodies, Monoclonal↗
PubMed2000

[Thirty years of activity at the center for cystic fibrosis in Parma].

One hundred fourteen patients (52 males, age range 1 to 40 yr, median age 16 yr) affected by Cystic Fibrosis (CF) are currently followed by the CF Centre of Parma. Forty two percent of them are adults. Most patients live in Emilia Romagna (59%) and Lombardia (19%). With respect to CF genotype, delta F508 mutation has been found in 54% of patients. Despite of the improvement in knowledge of CF, its diagnosis is still based on the clinical features and sweat test is usually used to confirm the diagnosis. Recent diagnostic tools, such as the genetic analysis and the potential difference measurement, could also be helpful in ambiguous situations. In the CF Centre of Parma, in agreement with the literature, the disease is now diagnosed earlier than in the past. Particularly, in the last year the median age of diagnosis was seven months, while it was 20 months in the 1980s. The early diagnosis and the improvement in the treatment of lung disease has significantly increased the median survival age of our patients, changing from 7.5 years in 1975 to 20 years in 1999. A significant improvement in CF prognosis could be further achieved by potential investigational agents, such as "protein-repair" addressed to CFTR activation. However, the gold standard for CF therapy is represented by gene therapy, although it is not yet available.

Adolescent↗
PubMed1985

Anatomic predisposition to perianal fistulae formation in the German shepherd dog.

Gross and microanatomic features which may predispose the German Shepherd Dog to perianal fistulae formation were studied in 2 groups of clinically healthy dogs: a predisposed group (German Shepherd Dogs) and a control group comprising breeds not ordinarily affected by perianal fistulae. The dimensions of the anal crypts (depth, base width, and length), measured and compared statistically between samples, identified no significant variation between groups (P greater than 0.05). Major tissue components of the anal canal were measured microscopically and were similarly evaluated: epithelial height in each zone, thickness of the lamina propria in each zone, thickness of the internal and external anal sphincter muscles, and density of the circumanal, sebaceous, and apocrine sweat glands. The only significant finding was an increase in density of apocrine sweat glands in the zona cutanea in the pre-disposed dog group. In a semiquantitative analysis of the inflammatory responses frequently seen in the anal glands, more mature fibroplasia was seen in the German Shepherd Dogs, indicating that inflammation was more longstanding in this group.

Animals↗
PubMed1990

Microcystic adnexal carcinoma. An immunohistochemical comparison with other cutaneous appendage tumors.

Since its initial description, microcystic adnexal carcinoma (MAC) of the skin has been controversial. In particular, it features keratin production of the type seen in some pilar neoplasms , and has been thought to pursue partial follicular differentiation. Diagnostically, MAC may be difficult to separate from desmoplastic trichoepithelioma (DTE) in superficial biopsy specimens. We studied 12 MACs, 22 malignant eccrine acrospiromas, 7 sudoriferous syringometaplasias, 6 syringomas, 5 DTEs, and 40 other benign pilar neoplasms immunohistochemically. Paraffin sections and antibodies to "hard" (pilar) keratins. epithelial membrane antigen (EMA), carcinoembryonic antigen (CEA), Leu-M1, and S100 protein were employed. The MACs exhibited reactivity for hard keratin subclasses AE 13 and AE 14, EMA, CEA, and Leu-M1. Desmoplastic trichoepitheliomas expressed positivity for AE 14, EMA, and Leu-M1 focally, but lacked the other specified markers. Syringomas and malignant acrospiromas displayed EMA, CEA, and AE 14 reactivity, and 5 syringometaplastic lesions were AE 14-reactive. Benign pilar tumors aside from DTEs were reactive only for AE 13, AE 14, or both. These data indicate that MAC exhibits an immunophenotype that is a "hybrid" of those seen in pure sweat glandular and follicular neoplasms, and suggest that it may indeed show combined pilar and sudoriferous differentiation. Based on these results, it also appears that immunohistochemical analysis may be useful in the diagnostic separation of MAC and DTE.

Adenoma, Sweat Gland↗
PubMed1993

Adenomyoepithelioma of the breast: fine-needle sampling with histologic, immunohistologic, and electron microscopic analysis.

Fine-needle sampling was performed in a woman with a subareolar breast mass. The cytologic diagnosis was consistent with a benign sweat gland-type tumor. Cytologic features included epithelial cells and spindle-shaped cells lying free or in fibrillary myxoid ground substance. Histologic study revealed the biphasic appearance of this tumor composed of proliferating myoepithelial cells and glandular epithelial cells as supported by immunohistologic and electron microscopic analyses. Epithelial cells were strongly positive for cytokeratins, and spindle cells were positive for actin, S-100 protein, and keratin and showed ultrastructurally typical features of myoepithelial cells.

Biopsy, Needle↗
PubMed2001

Endoscopic clipping in video-assisted thoracoscopic sympathetic blockade for axillary hyperhidrosis. An analysis of 26 cases.

BACKGROUND: Endoscopic thoracic sympathectomy or sympathicotomy is the standard method for the treatment of axillary hyperhidrosis. But postoperative compensatory sweating may be troublesome in some patients. Therefore, we use endoclips to perform the T3 and T4 sympathetic blockade instead of permanently interrupting the transmission of nerve impulses from the sympathetic trunk. METHODS: Between May 1997 and June 1998, a total of 26 patients with axillary hyperhidrosis underwent video-assisted thoracoscopic sympathetic blocking of the T3 and T4 ganglia at our hospital. There were 10 men and 16 women with a mean age of 31.7 years (range, 16-47). All patients were placed in a semi-sitting position under single-lumen intubated anesthesia. We performed the sympathetic blockade by clipping the T3 and T4 ganglia at the level of the third, fourth, and fifth rib beds using an 8-mm 0 degree thoracoscope. RESULTS: Bilateral T3 and T4 sympathetic blockade was achieved in all 26 patients. The operation was usually completed within 30 min (range, 20-42). Most patients were discharged within 4 h after the operation. Surgical complications were minimal, with only one case of segmental atelectasis (3.8%). There were no deaths. The mean postoperative follow-up period was 31.3 months (range, 24-37). Twenty-three patients (88.5%) developed compensatory sweating of the trunk and lower limbs. Twenty-four patients (92.3%) were satisfied with the results of the operation. Improvement of axillary hyperhidrosis was obtained in all patients. One patient underwent a reverse operation to remove the endoclips due to intolerable compensatory sweating; improvement was seen 25 days after removal of the clips. CONCLUSION: Video-assisted thoracoscopic T3 and T4 sympathetic blockade by clipping is a safe and effective method for the treatment of patients with axillary hyperhidrosis. Patients who experience excessive compensatory sweating may require a reverse operation for endoclip removal.

Adolescent↗
PubMed2004

Sweat gland morphology and periglandular innervation in essential palmar hyperhidrosis before and after treatment with intradermal botulinum toxin.

BACKGROUND: Intradermal botulinum toxin (Btx) produces long-lasting relief of focal hyperhidrosis, but its mechanism of action is poorly understood. OBJECTIVE: To study the effect of Btx A on the size and innervation of sweat glands in patients with palmar hyperhidrosis. METHODS: Palmar skin biopsy was performed in 26 hyperhidrotic patients before scheduled Btx treatment and in 11 controls. Twelve of the patients also underwent biopsy 1 to 6 months after the Btx injections. Sweat gland morphology was investigated by light microscopy; the cross-sectional area of the secretory tubule and its lumen was measured by image analysis. Immunofluorescence (IF) with antibodies to the neural markers protein gene product 9.5 (PGP 9.5) and growth-associated protein 43 (GAP 43), and to vasoactive intestinal peptide (VIP) and calcitonin gene-related peptide (CGRP), was used to analyze the periglandular innervation. RESULTS: The gross morphology of the sweat glands was similar in patients and controls, with no significant differences in tubular and luminal areas between the groups. After Btx treatment, the tubular dimensions remained unchanged, but the lumen tended to be smaller ( P = .07). Around the glands, increased GAP 43 staining indicating sprouting was seen within 3 months after Btx treatment ( P = .016); whereas the PGP 9.5 staining was decreased in most specimens ( P = .09) indicating lack of functional nerve growth. No change in VIP or CGRP immunoreactivity was observed. CONCLUSIONS: The sweat glands appear structurally normal in hyperhidrotic patients before Btx therapy, whereas after therapy the luminal area of the gland is frequently diminished. The IF data GAP 43/PGP 9.5 suggest that Btx therapy induces long-standing functional denervation of the sweat glands, which might explain its anti-transpiratory efficacy.

Adolescent↗
PubMed1996

Byler-like familial cholestasis in an extended kindred.

Progressive familial intrahepatic cholestasis (PFIC) occurs in many communities and races. A form of PFIC in five children from two consanguineous marriages in an Irish kindred is described. In addition, a review of clinical information from the records of three deceased members of the kindred strongly implies that they also suffered from PFIC. The children had a history of neonatal diarrhoea, sepsis, and intermittent jaundice that ultimately became permanent. They suffered intractable pruritus and growth retardation. Despite evidence of severe cholestasis, serum gamma-glutamyl transferase and cholesterol were normal in these children. Sweat sodium concentration were raised in three children. Liver histology showed severe intrahepatic cholestasis and hepatocellular injury. Urinary bile acid analysis revealed a non-specific pattern consistent with chronic cholestasis. These children suffer from a form of PFIC remarkably similar to that occurring in members of the Byler kindred.

Bile Acids and Salts↗
PubMed2001

Facial sweat gland carcinoma metastasizing to neck nodes: a diagnostic and therapeutic challenge.

We report an unusual case involving a patient with sweat gland carcinoma of the cheek who presented with ipsilateral neck lymph node metastasis 10 years after his initial presentation. Pathological analysis of the surgical specimen revealed a strong reactivity of tumor cells to gross cystic disease fluid protein 15, estrogen receptor protein, and progesterone receptor protein. On the basis of these results, tamoxifen citrate therapy was initiated empirically. Our patient has been disease free for more than 3 years. Based on this and another case reported in the literature, we believe that antiestrogen therapy could prove beneficial in a subset of patients with sweat gland carcinoma. We recommend future multicenter clinical trials to assess the effectiveness of postoperative tamoxifen therapy for patients with estrogen and progesterone receptor protein-positive metastatic sweat gland carcinoma.

Adenocarcinoma↗
PubMed2001

Reduction in immunosuppression as initial therapy for posttransplant lymphoproliferative disorder: analysis of prognostic variables and long-term follow-up of 42 adult patients.

BACKGROUND: Posttransplant lymphoproliferative disorder (PTLD) is an Epstein-Barr virus-associated malignancy that occurs in the setting of pharmacologic immunosuppression after organ transplantation. With the increased use of organ transplantation and intensive immunosuppression, this disease is becoming more common. We explore reduction in immunosuppression as an initial therapy for PTLD. METHODS: We analyzed our organ transplant patient database to identify patients with biopsy-proven PTLD who were initially treated with reduction of their immunosuppressive medications with or without surgical resection of all known disease. RESULTS: Forty-two adult patients were included in this study. Thirty patients were treated with reduction in immunosuppression alone. Twelve patients were treated with both reduction in immunosuppression and surgical resection of all known disease. Thirty-one of 42 patients (73.8%) achieved a complete remission. Of those patients who were treated with reduction in immunosuppression alone, 19 of 30 (63%) responded with a median time to documentation of response of 3.6 weeks. Multivariable analysis showed that elevated lactate dehydrogenase (LDH) ratio, organ dysfunction, and multi-organ involvement by PTLD were independent prognostic factors for lack of response to reduction in immunosuppression. In patients with none of these poor prognostic factors, 16 of 18 (89%) responded to reduction in immunosuppression in contrast to three of five (60%) with one risk factor and zero of seven (0%) with two to three factors present. The analysis also showed that increased age, elevated LDH ratio, severe organ dysfunction, presence of B symptoms (fever, night sweats, and weight loss), and multi-organ involvement by PTLD at the time of diagnosis are independent prognostic indicators for poor survival. With median follow-up of 147 weeks, 55% of patients are alive with 50% in complete remission. CONCLUSIONS: Reduction in immunosuppression is an effective initial therapy for PTLD. Clinical prognostic factors may allow clinicians to identify which patients are likely to respond to reduction in immunosuppression.

Adolescent↗
PubMed1989

Closed loop control of human body temperature: results from a one-dimensional model.

A one-dimensional model of human thermo-regulation is used to solve a variety of basic problems in determining an adequate structure of the controller for metabolic heat production, skin blood flow and sweat production. Assuming one integrated central and one integrated peripheral afferent signal the controller parameters are evaluated by analysis of the control performance. Based on a validation by experimental results this allows the determination of a first optimized set of values for controller gains and weight of skin temperature feedback. Furthermore we analyse the effect of inhomogeneous distribution of heat production and blood flow, the influence of body fat content, of controller gains, of weight of skin temperature feedback and of depth of peripheral receptors on the dynamic performance. Increase of peripheral blood flow in particular evokes essentially both an increase of energy requirement in the cold and a quicker system response. Differing rates of increase of metabolic heat production are the consequence of differing body fat content. The weight of skin temperature feedback can be limited to 5...20%, because values outside this range evoke dynamic responses incompatible with the experiments. The actual value can only be determined if there is a correct assumption for the depth of the skin receptors. The use of measured superficial skin temperatures brings about an underestimation of the peripheral afferent signal. Of the controller gains it is primarily the gain of the metabolic controller which affects the dynamic response of the system. The experimental fact of a delayed onset of sweat production after a transition from cold to heat is the consequence of a high gain of the vasomotor system.

Adipose Tissue↗
PubMed1999

Menopausal symptoms and symptom clustering in Chinese women.

OBJECTIVES: this paper aims to report the prevalence of symptoms in the Hong Kong Chinese perimenopausal women; to construct reported symptoms into symptom groupings; and to clarify whether the symptom groups are associated with menopausal status. METHODS: a random telephone survey of perimenopausal women aged 44-55 years was conducted in 1996. Eligible subjects were identified through telephone dialing of a random sample of the numbers listed in the residents' telephone directory. Standardized questionnaire, including a 22-item symptom check list, was administered over the telephone. The principal component analysis method followed by varimax rotation was used to examine the relations among the symptoms. RESULTS: differences in the prevalence of menstrual problems across the menopausal status were noted with perimenopausal women having the most complaints. Musculoskeletal conditions were the top complaints reported by the respondents, followed by headaches and psychological symptoms. About 10% of the women complained of hot flushes, and less than 5% of cold sweats. Five symptom clusters, namely psychological, musculoskeletal/gastrointestinal, non-specific somatic, respiratory, and vasomotor, have been identified. After adjustment for age, the analysis of variance showed that psychological, non-specific somatic and vasomotor symptoms were significantly associated with menopausal status, while musculoskeletal and respiratory were of borderline statistical significance. CONCLUSIONS: compared with pre- and post-menopausal women, perimenopausal women had the highest reports of symptom complaints. Musculoskeletal complaints were the most prevalent complaints, followed by psychological symptoms. While vasomotor symptoms were significantly associated with menopausal status, their prevalence was comparatively lower than that reported in Caucasian populations.

Adult↗
PubMed1990

New experimental and clinical data on the relationship between magnesium and sport.

Exercise under certain conditions appears to lead to Mg depletion and may worsen a state of deficiency when Mg intake is inadequate. Whereas hypermagnesaemia occurs following short term high intensity exercise as the consequence of a decrease in plasma volume and a shift of cellular magnesium resulting from acidosis, prolonged submaximal exercise is accompanied by hypomagnesaemia. Discordant findings on the effect of physical exercise on erythrocyte concentrations have been reported. A mechanism for the observed decrease in plasma magnesium concentration after long term physical exercise could be a shift of Mg into the erythrocyte. However, in several studies the decrease in plasma Mg was not accompanied by an increase in RBC Mg, but a decrease in cellular Mg was observed. Urinary Mg losses during an endurance event could play a role in this depletion but are often reduced, reflecting renal compensation. Loss of Mg by sweating takes place only when there is a failure in sweat homeostasis, a situation which arises when exercise is made in conditions of damp atmosphere and high temperature. Stress caused by physical exercise is capable of inducing Mg deficit by various mechanisms. A possible explanation for decreased plasma Mg concentration during long endurance events is the effect of lipolysis. Since fatty acids are mobilized for muscle energy, lipolysis would cause a decrease in plasma Mg. In developed countries Mg intake is often marginal and sport is a factor which is particularly likely to expose athletes to Mg deficit through metabolic depletion linked to exercise itself, which can only aggravate the consequences of a frequent marginal deficiency. Mg depletion and deficiency therefore play a role in the pathophysiology of physical exercise. Experiments on animals have shown that severe Mg deficiency reduces physical performance and in particular the efficiency of energy metabolism. These data, however, do not correspond to those of marginal deficiency most commonly observed in humans. Clinical symptomatology, both in athletes and in other patients, is dominated by the symptomatology of neuromuscular hyperexcitability. Medical authorities in sport have enforced obligatory tests for latent tetany in athletes, with ionic assessment. The effects of the correction of magnesium deficiency are judged from clinical signs, Chvosteck sign, electromyogram and echocardiogram findings and plasma Mg, erythrocyte and urine analysis. These may also be complemented by cardiac and respiratory investigations after exercise. The positive effects (analysis after a minimum period of one month) of a simple oral supplement administered in physiological doses (5 mg/kg body weight/day) provides evidence for the existence of a deficiency.(ABSTRACT TRUNCATED AT 400 WORDS)

Animals↗
PubMed1997

Sweat gland carcinoma. Case report and review of the literature.

BACKGROUND: We report a 57-year-old Hispanic man with malignant nodular hidradenoma. The patient had three biopsy-proven benign nodular hidradenomas over the left cheek over a 2-year period. A fourth recurrent lesion several months later had malignant histology. OBJECTIVE: Little is known about these rare sweat gland carcinomas and treatment modalities. We reviewed the literature on sweat gland carcinomas to elucidate the nature of these tumors and best treatment course. METHODS: Review of 76 published cases with compilation of data in tabular form with statistical analysis. RESULTS: The mean age was 57 years with equal male-to-female distribution. Tumor distribution is: lower extremities, 32.9%; upper extremities, 27.6%; trunk, 11.9%; head, 26.3%; neck, 1.3%. Metastatic sites are: lymph nodes, 30.2%; viscera, 22%. Malignant sweat gland neoplasms have a propensity for local and regional lymph node recurrence. The roles of lymph node dissection, radiation, and chemotherapy are reviewed. CONCLUSION: Treatment decisions should be formalized by conducting a well-structured trial.

Adenoma, Sweat Gland↗
PubMed2005

Combining immunoreactive trypsinogen and pancreatitis-associated protein assays, a method of newborn screening for cystic fibrosis that avoids DNA analysis.

OBJECTIVES: To evaluate the performance of a strategy in which, after immunoreactive trypsinogen (IRT) determination, genetic analysis is replaced by a biological test, the pancreatitis-associated protein (PAP) enzyme-linked immunosorbent assay (ELISA). STUDY DESIGN: The French newborn screening program includes cystic fibrosis (CF) screening by the IRT/CFTR mutation strategy. PAP was assayed on screening cards, in parallel with IRT, in all newborns from 5 French regions (n = 204,749). Analysis of PAP values in CF and non-CF newborns with elevated IRT allowed direct comparison between the current strategy and the proposed IRT/PAP strategy. RESULTS: A protocol in which newborns with IRT >50 ng/mL and PAP >1.8 ng/mL and those with IRT >100 ng/mL and PAP >1.0 ng/mL are directly recalled for sweat testing would have the same performance as the IRT/CFTR mutation strategy. CONCLUSIONS: The IRT/PAP strategy is an alternative for CF newborn screening, which avoids the drawbacks of genetic analysis and is cheaper and easier to implement than the current IRT/CFTR mutation strategy.

Antigens, Neoplasm↗
PubMed2006

Symptom prevalence and clustering of symptoms in people living with chronic hepatitis C infection.

Quality of life has been shown to be poor among people living with chronic hepatitis C. However, it is not clear how this relates to the presence of symptoms and their severity. The aim of this study was to describe the typology of a broad array of symptoms that were attributed to hepatitis C virus (HCV) infection. Phase 1 used qualitative methods to identify symptoms. In Phase 2, 188 treatment-naïve people living with HCV participated in a quantitative survey. The most prevalent symptom was physical tiredness (86%) followed by irritability (75%), depression (70%), mental tiredness (70%), and abdominal pain (68%). Temporal clustering of symptoms was reported in 62% of participants. Principal components analysis identified four symptom clusters: neuropsychiatric (mental tiredness, poor concentration, forgetfulness, depression, irritability, physical tiredness, and sleep problems); gastrointestinal (day sweats, nausea, food intolerance, night sweats, abdominal pain, poor appetite, and diarrhea); algesic (joint pain, muscle pain, and general body pain); and dysesthetic (noise sensitivity, light sensitivity, skin problems, and headaches). These data demonstrate that symptoms are prevalent in treatment-naïve people with HCV and support the hypothesis that symptom clustering occurs.

Adult↗