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PubMed1983

Metal-binding ligands in Viokase.

The object of this study was to detect low molecular weight zinc binders in Viokase, a porcine pancreatic preparation used in the treatment of variant acrodermatitis enteropathica (AE). Extracts were centrifuged and ultrafiltered to yield a soluble fraction containing solutes of molecular weights less than or equal to 500 daltons. These were studied by modified gel chromatography (MGC) using sealed Sephadex G-15-120 columns equilibrated with buffered solvent systems containing either 5 ppm copper (II) or 5 ppm zinc (II). Eluted fractions were assayed for these metals by atomic absorption and for amino acids by various techniques. MGC profiles of ultrafiltrates resulted in a peak consisting of glutamic acid and another peak consisting of several amino acids. A third peak was due to metal ions. Citrate and picolinate have been claimed to be important zinc-binding ligands in Viokase, but neither was detectable by MGC, although small amounts of citrate or similar compounds were detected by the Furth and Herrmann reaction. Since amino acid ligands are easily detectable by MGC, and metal distribution among ligands is competitive in nature, the possibility of a role for citrate or picolinate as a zinc transporter in Viokase is seriously reduced. Relief to the variant AE patient was probably not due to citrate- or picolinate-enhanced zinc uptake.

Acrodermatitis↗
PubMed1992

Impact of zinc supplementation on intestinal permeability in Bangladeshi children with acute diarrhoea and persistent diarrhoea syndrome.

Zinc has been shown to enhance intestinal mucosal repair in patients suffering from acrodermatitis enteropathica; but the impact on mucosal integrity during acute (AD) or persistent (PD) diarrhoea is unknown. One hundred eleven children with AD and 190 with PD aged between 3 and 24 months received, randomly and blind to the investigators, either an elemental zinc supplement of 5 mg/kg body wt/day or placebo in multivitamin syrup for 2 weeks while intestinal permeability and, biochemical and anthropometric markers were serially monitored. The permeability test was administered as an oral dose of 5 g lactulose/l g mannitol in a 20-ml solution followed by a 5-h urine collection. The ratio of the urinary probe sugars was correlated to clinical, biochemical, and microbiological parameters. At presentation, lactulose excretion was increased and mannitol excretion decreased in both AD and PD as compared with age-matched asymptomatic children. The lactulose/mannitol ratio (L/M) was higher in subjects with mucosal invasive pathogens (rotavirus and enteropathogenic Escherichia coli) compared with children excreting Vibrio cholera and enterotoxigenic E. coli. Two-week zinc supplementation significantly reduced lactulose excretion in both AD and PD, whereas the change in mannitol excretion and L/M was similar between study groups in both studies. Changes in lactulose excretion were significantly influenced by zinc supplementation in children with E. coli, Shigella sp., and Campylobacter jejuni stool isolates. The greatest reduction in total lactulose excretion was seen in supplemented children who on presentation were lighter (wt/age less than 80%), thinner (wt/ht less than 85%), and undernourished [middle upper arm circumference (MUAC) less than 12.5 cm] or with hypozincaemia (less than 14 mumol/L).(ABSTRACT TRUNCATED AT 250 WORDS)

Acrodermatitis↗
PubMed1986

Gianotti-Crosti syndrome: a study of 26 cases.

We have studied 26 patients presenting with a symmetrical papular or papulovesicular acrolocated eruption of more than 10 days duration. Mean age at onset was 2 years (range 10 months to 5.75 years). Lymphadenopathy was noted in eight cases, and hepatomegaly in one case. In 12 cases, histopathology and direct immunofluorescence were non-contributory. Cytolytic hepatitis occurred in one case and was associated with HBs antigenemia. A history of recent immunization was given in two cases. There was serological evidence of recent Epstein-Barr virus infection in seven out of 13 cases tested. Coxsackie B viruses were isolated from three patients, and cytomegalovirus was probably associated with the syndrome in one case. We conclude that the Gianotti-Crosti syndrome is not rare in France, and that non-hepatitis B virus (HBV)-associated cases are more frequent than the classical HBV-associated papular acrodermatitis of childhood.

Acrodermatitis↗
PubMed1990

Acral granulomatous dermatosis.

A 17-year-old boy developed an acral granulomatous dermatosis which resembled clinically acrodermatitis continua of Hallopeau. The histology showed dermal and subcutaneous abscess formation with granulomas. The lesions responded to systemic corticosteroid therapy, with residual atrophy and contractures of the fingers.

Acrodermatitis↗
PubMed1991

Periarticular fibrous nodules in Lyme borreliosis.

Periarticular fibrous nodules have been described in patients with late-phase acrodermatitis chronica atrophicans (ACA) in the setting of Lyme borreliosis. We report the clinical and histopathological study of two patients with periarticular fibrous nodules and with Lyme borreliosis and discuss the relationship between these nodules and ACA.

Acrodermatitis↗
PubMedDate not supplied

Human leukocyte antigen in genodermatosis.

Tissue typing performed on the lymphocytes of 41 patients, including: 23 patients with ichthyosis (15 of autosomal dominant and 8 of the x-linked variety; 8 patients with tuberous sclerosis (epiloia); and 10 patients with acrodermatitis enteropathica (AEP). In addition, tissue typing was performed for a control group of 80 healthy unrelated subjects of the same ethnic origin as the patients. A significant increase of A2 antigen or its crossreacting antigen A28 was detected among the three diseases studied. A significant increase of HLA-A2 antigen was found in autosomal dominant ichthyosis, while the x-linked variety showed significant association with HLA-Cw2. A significant association with HLA-A28 antigen was noticed in both epiloia and AEP. The suggested mechanisms of such association may be referred to as incidental genetic linkage with mutants causing disease or deficiency.

Acrodermatitis↗
PubMed1995

Transient zinc deficiency in a breast-fed premature infant.

Symptomatic zinc deficiency developed in a breast-fed premature male infant of 31 weeks gestation. At 13 weeks of age he presented with diarrhoea, irritability and an eruption identical to acrodermatitis enteropathica. Breast milk zinc concentrations were low. His course was complicated by milk protein intolerance. After 7 weeks, zinc supplementation was ceased without recurrence of disease.

Acrodermatitis↗
PubMed1992

Acquired zinc deficiency in association with anorexia nervosa: case report and review of the literature.

Zinc deficiency, whether a result of an acquired or inherited abnormality of zinc metabolism, is associated with characteristic cutaneous findings. The inherited variety is known as acrodermatitis enteropathica. We present a case of zinc deficiency secondary to starvation induced by anorexia nervosa. Since the cutaneous stigmata of zinc deficiency and anorexia nervosa can initially be subtle and occasionally overlap, we believe that screening zinc levels in patients with anorexia nervosa with prominent cutaneous findings should be considered.

Acrodermatitis↗
PubMed1975

Recalcitrant pustular eruptions of the extremities.

Forty-seven biopsies of pustules from patients with recalcitrant pustular eruptions of the palms and soles were examined blind and the results were subsequently compared among three clinical sub-groups: acrodermatitis continua, pustular psoriasis and pustulosis palmaris et plantaris. With the exception of three cases, it was not possible to make a diagnosis of psoriasis on the basis of histology alone. Histological findings were otherwise uniform and typical: intra-epidermal, unilocular, well-delineated pustules. No significant differences were found among the three subgroups. In view of the uniform histology, the unknown etiology and the uncertain clinical classification, the authors prefer the clearly descriptive term pustulosis palmaris et plantaris.

Acrodermatitis↗
PubMed1977

Selenium content of tissues in Finnish infants and adults with various diseases, and studies on the effects of selenium supplementation in neuronal ceroid lipofuscinosis patients.

A low blood selenium level has previously been observed in healthy inhabitants of Finland (WESTERMARCK et al. 1977). In this study even lower blood selenium values were observed in patients with acrodermatitis enteropathica, dystrophia musculorum progressiva (Duchenne), infantile and juvenile type of neuronal ceroid lipofuscinosis (NCL), severe mental retardation caused by various factors, and myocardial infarction. The selenium content of the brain, heart, kidney and liver in patients of different ages was also determined. The highest selenium level was found in the kidney. The mean liver selenium concentrations in stillborn, premature and full-term neonates were 1.11 +/- 0.23 (8), 1.21 +/- 0.17 (12) and 0.93 +/- 0.16 microgram/g dry weight (12) respectively (the number of subjects in parentheses). The selenium values are considerably higher than those in infants of from one to nine months of age and adults, whose liver selenium values were 0.58 +/- 0.21 (8) and 0.67 +/- 0.08 microgram/g dry weight (8) respectively. The vitamin E levels of serum in patients with NCL, as well as in subjects with severe mental retardation (controls), were low compared with values in healthy normal subjects. Sodium selenite supplementation in patients with NCL produced at least a transitory improvement without causing any toxic effects during one year of administration.

Acrodermatitis↗
PubMed1977

Zinc binding: a difference between human and bovine milk.

Gel chromatography indicated that most of the zinc in cow's milk was associated with high-molecular-weight fractions, whereas zinc in human milk was associated with low-molecular-weight fractions. A species difference in zinc-binding ligands may explain why symptoms of the genetic disorder of zinc metabolism, acrodermatitis enteropathica, can be alleviated by feeding human but not cow's milk.

Acrodermatitis↗
PubMed1986

Of mice and men, metals and mutations.

Several mutations affecting the transport of copper and zinc in humans and in mice have been discovered over the last 15 years, joining the long known disturbance of copper transport in Wilson's disease. Menkes' disease (classical and mild variant forms) and X linked Ehlers-Danlos syndrome (type IX, X linked cutis laxa) have features in common with one another and with the brindled (Mobr) and blotchy (Moblo) mouse mutants, respectively. There may be one allelic series of mutants in each species or two loci may be involved in each. The toxic milk mutant (tx) in the mouse may be homologous to Wilson's disease in man. The defect of intestinal absorption of zinc in acrodermatitis enteropathica has no homologue yet in the mouse. However, the lethal milk (lm) mutant in the mouse may be homologous to a condition of zinc deficiency described in a few breastfed, low birth weight infants. Many more genetic defects of transport of copper and of zinc may await discovery. Conversely, these mutants are valuable in elucidating the normal processes of copper and zinc transport.

Acrodermatitis↗
PubMed1985

Clinical manifestations of zinc deficiency.

The essentiality of zinc for humans was recognized in the early 1960s. The causes of zinc deficiency include malnutrition, alcoholism, malabsorption, extensive burns, chronic debilitating disorders, chronic renal diseases, following uses of certain drugs such as penicillamine for Wilson's disease and diuretics in some cases, and genetic disorders such as acrodermatitis enteropathica and sickle cell disease. In pregnancy and during periods of growth the requirement of zinc is increased. The clinical manifestations in severe cases of zinc deficiency include bullous-pustular dermatitis, alopecia, diarrhea, emotional disorder, weight loss, intercurrent infections, hypogonadism in males; it is fatal if unrecognized and untreated. A moderate deficiency of zinc is characterized by growth retardation and delayed puberty in adolescents, hypogonadism in males, rough skin, poor appetite, mental lethargy, delayed wound healing, taste abnormalities, and abnormal dark adaptation. In mild cases of zinc deficiency in human subjects, we have observed oligospermia, slight weight loss, and hyperammonemia. Zinc is a growth factor. Its deficiency adversely affects growth in many animal species and humans. Inasmuch as zinc is needed for protein and DNA synthesis and for cell division, it is believed that the growth effect of zinc is related to its effect on protein synthesis. Whether or not zinc is required for the metabolism of somatomedin needs to be investigated in the future. Testicular functions are affected adversely as a result of zinc deficiency in both humans and experimental animals. This effect of zinc is at the end organ level; the hypothalamic-pituitary axis is intact in zinc-deficient subjects. Inasmuch as zinc is intimately involved in cell division, its deficiency may adversely affect testicular size and thus affect its functions. Zinc is required for the functions of several enzymes and whether or not it has an enzymatic role in steroidogenesis is not known at present. Thymopoeitin, a hormone needed for T-cell maturation, has also been shown to be zinc dependent. Zinc deficiency affects T-cell functions and chemotaxis adversely. Disorders of cell-mediated immune functions are commonly observed in patients with zinc deficiency. Zinc is beneficial for wound healing in zinc-deficient subjects. In certain zinc-deficient subjects, abnormal taste and abnormal dark adaptation have been noted to reverse with zinc supplementation.

Acrodermatitis↗
PubMed1978

Intestinal absorption of zinc: a role for a zinc-binding ligand in milk.

This study examined the proposal that a low molecular weight, zinc-binding ligand (ZBL) in certain milks is important for zinc absorption in the neonatal period. Ten-day-old rats, in which intestinal ZBL is absent, fed (by stomach intubation) 65Zn-labeled ZBL from rat milk, absorbed significantly more 65Zn than those fed free 65ZnCl2 or bovine milk fractions. ZBL from human milk appeared to have an intermediate effect, possibly due to species specificity. 65Zn was found in the ZBL fraction in intestinal mucosa of 10-day-old rats fed rat or human milk fractions, but not in those fed bovine milk or free 65ZnCl2. In contrast, in 18-day-old rats, which have an endogenous intestinal ZBL, there were no differences in zinc absorption, and any of the labeled milk fractions or free 65Zn caused localization of 65Zn in the ZBL fraction of intestinal mucosa. These results support the hypothesis that the intestinal ZBL plays a role in zinc absorption and that in the neonatal period before its development the milk ZBL is valuable for this function. This mechanism may be important in normal human infants as well as in acrodermatitis enteropathica patients.

Acrodermatitis↗
PubMedDate not supplied

Zinc deficiency and lymphocyte subpopulations. A study by flow cytometry.

Zinc deficiency is well known to alter immunity. We report the case of a 18-yr-old female with relapsing Crohn's disease who experienced acrodermatitis enteropathica due to zinc deficiency during total parenteral nutrition (TPN). Blood lymphocytes have been studied by flow cytometry: before zinc treatment an important decrease of T-helper lymphocytes with high level of OKM-5+ lymphocytes had been observed. Zinc-supplemented diet induced within a few days, a rise of T-helper lymphocytes and a proportional reduction of OKM5+ cells. Increased values of high metabolism surface marker (OKT-9) were also observed, as well as cytoplasmic modifications. The authors suggest that lymphocyte surface markers could be useful to monitor TPN in patients at high risk for zinc deficiency.

Acrodermatitis↗
PubMed1996

Bovine hereditary zinc deficiency: lethal trait A 46.

Bovine hereditary zinc deficiency, also referred to as Adema disease, is an autosomal recessive disorder which results in inadequate amounts of zinc being absorbed from the gastrointestinal tract and leads to a number of clinical abnormalities. Using semen from a homozygous affected bull and obligate heterozygote cows in embryo transfer studies, 7 offspring were obtained. These included 5 affected calves and 1 heterozygous carrier; the seventh calf died within 48 hours of birth undiagnosed. One unaffected, unrelated bull calf was raised as a control. All the calves were raised and maintained under similar management conditions designed to minimize secondary complications that would obscure the clinical and biochemical observations of a zinc deficient state. The first clinical manifestation of zinc deficiency was diarrhea, followed by skin lesions, poliosis, and a decreased ability to sustain a suckle reflex. Trace mineral analysis of plasma blood samples revealed that plasma zinc concentrations of all the calves were normal at birth; however, they gradually declined in affected calves over the course of 3-8 weeks postpartum to below 0.5 ppm. Biochemical analysis of serum samples showed alkaline phosphatase activity consistently paralleled changes in the plasma zinc concentrations. The oral administration of zinc acetate caused a reversal of all clinical, biochemical, and histologic abnormalities in affected calves. The study of these affected calves allows further insight into the biological role of zinc as well as provides an animal model for the continued investigation of the human homologue acrodermatitis enteropathica.

Acrodermatitis↗
PubMed2007

Focus floating microscopy: "gold standard" for cutaneous borreliosis?

Borrelia burgdorferi is difficult to detect in routine biopsy material from patients with skin lesions of borreliosis. In this study, a new immunohistochemical method, focus floating microscopy (FFM), was developed to detect B burgdorferi in tissue sections and was compared with polymerase chain reaction (PCR). By using standard histologic equipment, tissue sections stained with a polyclonal B burgdorferi antibody were simultaneously scanned through 2 planes: horizontally in serpentines and vertically by focusing through the thickness of the section. Borrelia were detected in 47 of 71 ticks, 34 of 66 tick bites, 30 of 32 erythema chronicum migrans cases, 41 of 43 borrelial lymphocytomas, and 50 of 51 acrodermatitis chronica atrophicans cases. FFM proved to be more sensitive than PCR (96.0% vs 45.2%) and nearly equally specific (99.4% vs 100%). All 169 control cases, except 1 false-positive case of secondary syphilis, were negative with FFM. FFM is an easy, quick, and inexpensive method to reliably detect Borrelia in cutaneous tissue sections.

Acrodermatitis↗
PubMed1983

Ultrastructural alterations of Paneth cells in infants associated with gastrointestinal symptoms.

Paneth cells containing abundant and large inclusion bodies found in patients with acrodermatitis enteropathica have been considered as a consistent and characteristic abnormality of this disease. We examined 34 children with gastrointestinal symptoms due to a wide variety of basic diseases and found similar abnormal Paneth cells in most of them, particularly in infants under the age of one year. This abnormality, according to our findings, is attributed to the secondary effect of zinc deficiency due to malnutrition.

Acrodermatitis↗