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PubMed1975

Zinc therapy of acrodermatitis enteropathica.

The therapeutic effect of orally administered zinc was evaluated in an adult woman with acrodermatitis enteropathica. When she was off therapy and in clinical relapse the plasma zinc concentration (10 mug per 100 ml), serum alkaline phosphatase (3 1U per liter) and urine zinc excretion rate (39 mug per 24 hours) were extremely low. Di-iodohydroxyquin therapy was accompanied by a modest increase in plasma zinc concentrations. Oral zinc sulfate (220 mg three times a day or 50 mg twice a day) resulted in rapid and complete clinical remission, and in a return of plasma zinc, serum alkaline phosphatase and urinary zinc excretion to normal. These data are compatible with a severe zinc deficiency state and indicate that the inherited defect in this disease is either in or closely related to zinc metabolism. The beneficial effects of zinc therapy in this patient provide further confirmation of the efficacy of oral zinc in the treatment of acrodermatitis enteropathica.

Acrodermatitis↗
PubMed2003

The acrodermatitis enteropathica gene ZIP4 encodes a tissue-specific, zinc-regulated zinc transporter in mice.

The human ZIP4 gene (SLC39A4) is a candidate for the genetic disorder of zinc metabolism acrodermatitis enteropathica. To understand its role in zinc homeostasis, we examined the function and expression of mouse ZIP4. This gene encodes a well conserved eight-transmembrane protein that can specifically increase the influx of zinc into transfected cells. Expression of this gene is robust in tissues involved in nutrient uptake, such as the intestines and embryonic visceral yolk sac, and is dynamically regulated by zinc. Dietary zinc deficiency causes a marked increase in the accumulation of ZIP4 mRNA in these tissues, whereas injection of zinc or increasing zinc content of the diet rapidly reduces its abundance. Zinc can also regulate the accumulation of ZIP4 protein at the apical surface of enterocytes and visceral endoderm cells. These results provide compelling evidence that ZIP4 is a zinc transporter that plays an important role in zinc homeostasis, a process that is defective in acrodermatitis enteropathica in humans.

Acrodermatitis↗
PubMed1983

The effects of diet and zinc treatment on the fatty acid composition of serum lipids and adipose tissue and on serum lipoproteins in two adolescent patients with acrodermatitis enteropathica.

In two adolescent patients with acrodermatitis enteropathica fatty acid spectra in serum lipids and adipose tissue and serum lipoprotein concentrations were followed for about 7 yr. One patient was treated by diet and iv infusions of high amounts of linoleic acid and later by different doses of zinc. The other boy was given only varying doses of zinc. Extra supply of linoleic acid raised its concentrations in serum triglycerides, cholesterol esters, phospholipids, and adipose tissue lipids from low to normal or high levels. In both patients linoleic acid in serum lipids was sensible to the dose of zinc, decreasing when it was low and increasing when it was high. Serum triglycerides increased when the supply was low and was normalized when high doses were given. High-density lipoprotein cholesterol, however, remained low throughout the study. We conclude that in acrodermatitis enteropathica zinc thus seems to be of importance in regulating linoleic acid and serum lipoprotein metabolism.

Acrodermatitis↗
PubMed1996

Etiology of the acrodermatitis chronica atrophicans lesion in Lyme disease.

Spirochete diversity in acrodermatitis chronica atrophicans lesions in a closely defined central European site was compared to that in the local vector population, in human erythema migrans lesions, and in cerebrospinal fluid by amplifying and sequencing a segment of the gene of outer surface protein A directly from sampled tissues. Borrelia garinii, Borrelia afzelii, and Borrelia burgdorferi acutely infect human skin and invade internal tissues. Only B. afzelii, however, is associated with acrodermatitis chronica atrophicans lesions, persisting chronically where the skin has atrophied.

Acrodermatitis↗
PubMed1996

The acrodermatitis enteropathica mutation transiently affects zinc metabolism in human fibroblasts.

The acrodermatitis enteropathica (AE) mutation has been shown to affect zinc transport in human intestinal biopsies. However, whether the mutation is also expressed in human fibroblasts has not been determined. The activity of the zinc-dependent enzyme, 5' nucleotidase, and cell zinc content were measured in normal and AE fibroblasts 2 and 4 d after subculturing to determine the effect of the AE mutation on zinc metabolism. The activity of 5' nucleotidase in AE cells was 68% of normal at 2 d after subculturing. Although 5' nucleotidase activity had decreased significantly in both normal and AE fibroblasts at 4 d after subculturing, there was no significant difference between the two genotypes. The zinc content of AE fibroblasts was also significantly reduced. Acrodermatitis enteropathica fibroblasts contained 62% less zinc than normal fibroblasts at 2 d. By 4 d the normal fibroblast zinc content had decreased to that of the AE fibroblasts. The uptake and transport of 65Zn into AE fibroblasts at 2 d was measured because these cells exhibited reduced 5' nucleotidase activity and cell zinc content at this time. The uptake of zinc over a 90-min time period was the same in the two genotypes. However, AE fibroblasts incubated with 2-10 mumol Zn/L for 15 min had significantly slower zinc transport compared with normal fibroblasts. In both genotypes, Michaelis-Menten kinetics were observed. Normal and AE fibroblasts had similar affinities for zinc (Km), but AE fibroblasts exhibited a Vmax which was reduced by 38%. These results indicate that the phenotypic expression of the AE mutation occurs in a time-dependent manner, is not restricted to the intestine and is also transiently expressed in human fibroblasts, resulting in abnormal zinc metabolism in these cells.

5'-Nucleotidase↗
PubMed2004

Edema, anemia, hypoproteinemia, and acrodermatitis enteropathica: an uncommon initial presentation of cystic fibrosis.

Cystic fibrosis is a genetic disorder characterized by chronic obstructive pulmonary disease, pancreatic exocrine deficiency, and abnormally high sweat electrolyte concentrations. Less frequently, the presenting features in infants may include edema, anemia, hypoproteinemia, and acrodermatitis enteropathica. Liver involvement may produce hepatomegaly and mild elevation of transaminases. This clinical symptom usually presents within the first 6 months of life and is associated with a high morbidity and mortality. Early recognition and institution of appropriate nutritional supplementation and pancreatic enzymes is essential to improve outcome. Since the sweat test may be falsely negative, emergency physicians must maintain a high index of suspicion to make the diagnosis of cystic fibrosis in an infant who presents with edema, anemia, hypoproteinemia, and acrodermatitis enteropathica.

Acrodermatitis↗
PubMed2000

A case of acrodermatitis enteropathica.

Acrodermatitis enteropathica is a rare hereditary disorder affecting zinc metabolism that is characterized by dermatitis, alopecia, gastrointestinal disturbances, eye infections, and growth failure. We report a 17-month-old girl with acrodermatitis enteropathica. Physical examination showed a cutaneous eruption consisting of vesiculobullous and psoriasiform skin lesions symmetrically distributed in the perioral, acral, and perineal areas. Her plasma zinc level was decreased (75 micrograms/dl), but within the normal range (60.00-135.00 micrograms/dl). The patient was given zinc sulfate 50 mg/day. At the end of two months, she had significantly improved.

Acrodermatitis↗
PubMed1988

Self-limiting acrodermatitis enteropathica. A follow-up study of three interrelated families.

A variant of acrodermatitis enteropathica is described that has its onset before weaning and clears when the child starts its normal solid diet. A pedigree with three interrelated families is reported where 10 children were afflicted with this variant. They had symptoms of hypozincemia for a brief period during infancy. At the time of this study, they were symptom-free and their serum zinc levels were found to be within normal limits. The term "self-limiting acrodermatitis enteropathica" is proposed for the variant. In one lactating mother, the mammary zinc secretion was determined and was found to be deficient and unresponsive to oral zinc supplements. The possible mode of inheritance is also discussed.

Acrodermatitis↗
PubMedDate not supplied

Acrodermatitis enteropathica-like eruptions in a child with Hartnup disease.

Acrodermatitis enteropathica-like eruptions, not related to zinc deficiency, have been rarely reported in some metabolic disorders. Reported patients usually had low levels of essential amino acids, particularly isoleucine. Here we report a girl who first presented with an acrodermatitis enteropathica-like eruption and eventually had the diagnosis of Hartnup disease with a normal isoleucine level. We discuss the probable cause of her skin lesions and the differential diagnosis with pellagra.

Acrodermatitis↗
PubMed1976

Hypertension in a patient with acrodermatitis enteropathica.

One patient with acrodermatitis enteropathica complicated by hypertension is presented. The pathogenesis of the markedly elevated blood pressure is unknown. The acrodermatitis healed completely on zinc therapy and parallel to this improvement the hypertension could easily be controlled with a gradually decreased antihypertensive drug dosage.

Acrodermatitis↗
PubMed1981

Acrodermatitis enteropathica Reversibility of cerebral atrophy with zinc therapy.

A six-month-old Saudi boy with acrodermatitis enteropathica confirmed by low serum zinc and alkaline phosphatase levels is described. Both the patient and a sibling developed acrodermatitis enteropathica while entirely breastfed. The mother had low serum zinc levels. Cranial computed tomography initially showed marked central and cortical "atrophy" that improved on treatment with zinc sulphate. The importance of zinc for normal brain growth and function is well known and the improvement on cranial computed tomography could be explained by improved myelination.

Acrodermatitis↗
PubMed1975

Peripheral neuropathy in acrodermatitis chronica atrophicans (Herxheimer).

Acrodermatitis chronica atrophicans is a dermatological condition that takes a chronically progressive course and finally leads to a widespread atrophy of the skin. Involvement of the peripheral nervous system is frequently observed, predominantly a sensory polyneuropathy. General reactions, the effect of penicillin treatment, the histological findings, and reports concerning a communicable agent transmittable from human to human as well in tissue cultures point to an infectious disease. Acrodermatitis chronica atrophicans follows a peculiar geographical distribution forming clusters of high prevalence in certain regions. Transmission by ticks is suggested.

Acrodermatitis↗
PubMed1996

Acrodermatitis enteropathica secondary to Crohn's disease.

A patient suffering from Crohn's disease (CD) presented with alopecia, eczematoid and psoriasiform lesions located on the extremities, around the orifices and at pressure points, suggesting acrodermatitis enteropathica. Lately, her inflammatory bowel disease had flared up with abdominal pain, diarrhea and weight loss related to a retroperitoneal abscess. Acrodermatitis enteropathica due to zinc deficiency is a well-known complication of CD. In our patient the etiological factors involved appeared to be multiple.

Acrodermatitis↗
PubMed1976

Acrodermatitis enteropathica-like eruption association with parenteral nutrition.

This is a case report of a patient with acrodermatitis enteropathica-like eruption that occurred during parenteral nutrition. The skin lesions were similar to those of acrodermatitis enteropathica, but there were no other major symptoms such as diarrhea and alopecia. It was noticed that the lack of linoleic acid in the diet might be the cause of lesions.

Acrodermatitis↗
PubMed1983

The effect of zinc therapy on lysosomal inclusion bodies in intestinal epithelial cells in acrodermatitis enteropathica.

Two patients with acrodermatitis enteropathica had abnormal lysosomal inclusion bodies in the intestinal epithelial cells while in relapse but fewer smaller ones after zinc therapy. The third patient with acrodermatitis enteropathica in remission on zinc therapy had no inclusion bodies. The smaller inclusion bodies were similar to those found in coeliac disease.

Acrodermatitis↗
PubMed1994

Changes in Borrelia burgdorferi-specific serum IgG antibody levels in patients treated for acrodermatitis chronica atrophicans.

The kinetics of Borrelia burgdorferi-specific serum IgG antibody values in 74 patients treated for acrodermatitis chronica atrophicans was analysed by means of enzyme-linked immunosorbent assay. At the last clinical control, there had been no clinical signs of active infection. The serological follow-up time ranged from 12 months to 5 1/2 years (median 2 years and 1 month). In 68 (92%) of the 74 patients, a significant decrease of the specific antibody values was found within 3 years after the initiation of therapy. In 53 (72%) of the patients, this decrease was found within 15 months. Most of the patients remained seropositive during the follow-up period. The results show that a significant decline of the levels of serum IgG antibodies to Borrelia burgdorferi can be expected in the majority of patients who do not exhibit clinical evidence of persistent infection after antibiotic treatment of acrodermatitis chronica atrophicans.

Acrodermatitis↗
PubMed1994

Serological follow-up after treatment of Borrelia arthritis and acrodermatitis chronica atrophicans.

To study the serological response to Borrelia burgdorferi after treatment of late Lyme borreliosis, consecutive serum samples from 20 patients with Borrelia arthritis and 21 with acrodermatitis chronica atrophicans were analysed with capture IgM ELISA and indirect IgG ELISA, both using B. burgdorferi flagella as antigen. Seven patients had positive IgM OD values, whereas all 41 had positive IgG OD values before therapy. In the majority, highly elevated IgG OD values were seen. All patients improved after antibiotic therapy, 32 recovering completely, while 9 had sequelae. At follow-up after 6 months to 5 years, 4/7 patients became negative IgM ELISA, whereas 3 still had slightly elevated IgM OD values 6 months, 1 year and 4.5 years, respectively, after therapy. Only one patient became negative in IgG ELISA during follow-up, although a significant decline in IgG OD values was seen in 22 of the remaining 40 initially IgG-positive patients. The serological response after successful treatment of Borrelia arthritis and acrodermatitis chronica atrophicans may persist for several years even with highly elevated IgG OD values in patients who have recovered completely.

Acrodermatitis↗
PubMed1981

Clioquinol intoxication occurring in the treatment of acrodermatitis enteropathica with reference to SMON outside of Japan.

Many past cases of SMON have occurred in Japan but since 1975 no new cases have been reported. One type of SMON is associated with acrodermatitis enteropathica which has a very high frequency of occurrence in association with administration of clioquinol. Acrodermatitis enteropathica with SMON almost always has a high occurrence of visual impairment, but other sensory disturbances are slight or absent, while common adult SMON is just the opposite. We do not know why these differences occur.

Acrodermatitis↗