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At least 973 records · Page 54Linked to original sources
PubMed1997

Phacomatosis pigmentokeratotica: a patient with the rare melanocytic-epidermal twin nevus syndrome.

We describe a 10-year-old girl affected with a speckled lentiginous nevus and an epidermal nevus of the organoid type on corresponding parts of the body. On histopathological examination, the lesions showed epidermal hyperpigmentation and melanocytic hyperplasia on the one hand and verrucous epidermal acanthosis with sebaceous hyperplasia on the other hand. Except for a minor deviation of the spine, the patient had no obvious extracutaneous symptoms. Happle et al. have recently interpreted the rare co-occurrence of these two types of nevi in spatial proximity as an example of twin spotting in human skin and proposed the name 'phacomatosis pigmentokeratotica'. In most cases, additional skeletal or neurological anomalies are found. These are dissimilar from the extracutaneous symptoms of the sebaceous nevus syndrome, from which phacomatosis pigmentokeratotica should be distinguished. Molecular studies are needed to prove the concept of twin spotting and to reveal a link to the extracutaneous manifestations.

Child↗
PubMed1997

Low-dose danazol in the treatment of livedoid vasculitis.

BACKGROUND: Livedoid vasculitis is characterized clinically by smooth or depressed ivory-white scars surrounded by hyperpigmentation and telangiectasia with or without preceding purpuric infiltrated papules and plaques and histologically by intravascular deposition of fibrin. Its etiology remains obscure and therapy very difficult. OBJECTIVE: Our purpose was to test the efficacy of low-dose danazol in the treatment of livedoid vasculitis. METHODS: Seven patients with active lesions of livedoid vasculitis were treated with low-dose danazol (200 mg, orally, daily). Laboratory coagulation and fibrinolysis parameters, including antithrombin III, protein C, protein S, tissue plasminogen activator, plasminogen, alpha 2-antiplasmin and fibrinogen, were evaluated before and during the therapy. RESULTS: Six of the 7 patients completed the treatment. After the therapy, all 6 patients had rapid cessation of new lesion formation, prompt reduction in their pain and healing of ulcers. A significant elevation of plasminogen and a decrease in fibrinogen levels were noted 1 month after initiation of the therapy (p = 0.028). The level of fibrinogen seemed to parallel the disease activity in individual patients. In addition, in most of these patients, the levels of antithrombin III, protein C, protein S and alpha 2-antiplasmin tended to increase after the treatment. However, the differences were not statistically significant. Abnormalities of tissue plasminogen activator levels were less consistent. Low-dose danazol was well tolerated without major side effects. CONCLUSION: We concluded that low-dose danazol was effective in the treatment of livedoid vasculitis, without unacceptable side effects.

Administration, Oral↗
PubMed1995

Recurrent inflammation in incontinentia pigmenti of a seven-year-old child.

A young girl has undergone the typical skin manifestations of incontinentia pigmenti, leaving now only discrete hyperpigmentation on the flexor sides of the lower extremities. Each time when she has a feverish infection, a recurrence of inflammation can be seen within the hyperpigmented areas. At the same time when the fever subsides, the acute skin manifestations disappear as well. The combination of recurrent inflammatory skin manifestations within the hyperpigmented skin areas of incontinentia pigmenti and fever is unique. The reasons for this phenomenon and for its strict limitation to the areas of incontinentia pigmenti remain unclear.

Child↗
PubMed1994

Brown papules and leukoderma in Darier's disease: clinical and histological features.

We report a Japanese case of Darier's disease with brown and white papules or maculae distributed on the neck, trunk and dorsa of hands. Both brown papules and leukoderma showed typical histological features of Darier's disease. Moreover, there were much fewer melanocytes and melanosomes in the epidermis of both lesions. The corneal layer of the brown papules was far thicker than that of the leukoderma. Therefore, the thick corneal layer of the brown papules may prolong the retention of a few melanosomes or a little melanin to induce hyperpigmentation, while the thin corneal layer of leukoderma may not do so, thereby producing hypopigmentation. The difference in clinical and histological courses after involvement of melanocytes was proposed to be the cause of the discrepancy between the previous reports. We conclude that leukoderma was the primary lesion, the postinflammatory depigmented spots, or the atypical or subclinical eruption of Darier's disease.

Adult↗
PubMed1996

Systematic inspection of insulin injection sites for local complications related to incorrect injection technique.

We systematically inspected insulin injection sites in 100 insulin-requiring patients attending the Diabetic Clinic of the Tikur Anbassa Hospital (TAH) in order to identify local complications related to incorrect injection technique: local complications were found in 53 cases: skin hyperpigmentation and/or indurations in 30 patients; and fat atrophy or hypertrophy in 31 patients. Illiteracy was significantly more common among those with local complications (18/53 versus 6/47, chi 2 5.03, P < 0.05). Mean fasting blood glucose on the day of the inspection was significantly higher (14.9 + 6.3 mmol/l versus 10.5 + 6.1 mmol/l, P < 0.001) and a fasting blood glucose > 10 mmol/l more common (41/53 versus 20/47, chi 2 14.1, P < 0.0005) in those with than in those without local complications. There was no significant difference between the two groups in mean duration of diabetes (6.9 + 5.6 years versus 6.6 + 5.8 years), frequency of hypoglycaemic episodes (12/53 versus 5/47, chi 2 1.76, P > 0.05) or mean daily insulin dose (44 + 18 units versus 44 + 22 units per day). Therefore, we concluded that local complications resulting from incorrect injection technique, a common finding in the group of patients studied, may be common among insulin requiring diabetic patients in general. Incorrect insulin injection causes local complications and disfigurement which may compromise compliance. Furthermore, insulin absorption tends to be erratic from intradermal and fat hypertrophy sites thus interfering with effective diabetic control. Insulin injection sites should be inspected routinely to detect and correct family technique promptly.

Adipose Tissue↗
PubMedDate not supplied

Syndrome of congenital adrenocortical unresponsiveness to ACTH. Report of six patients.

Familial glucocorticoid deficiency (FGD) or unresponsiveness to ACTH at the receptor level is a rare autosomal recessive hereditary syndrome characterized by a low cortisol level despite high serum ACTH concentration. Aldosterone levels are normal. The clinical entity generally presents in the first year of life with skin hyperpigmentation and hypoglycemic convulsions. Cortisol response to exogenous ACTH is also absent. Unresponsiveness to ACTH may be due to a mutation in the ACTH receptor; sometimes no mutation is found. We discuss the clinical and laboratory findings and genetic studies in six patients with a diagnosis of FGD. A homozygous V142L mutation was detected in three of the patients and a homozygous D103N mutation was detected in two patients.

Adrenal Cortex↗
PubMed2001

Drug-induced skin pigmentation. Epidemiology, diagnosis and treatment.

Drug-induced pigmentation represents 10 to 20% of all cases of acquired hyperpigmentation and this hypothesis must be systematically raised in unexplained pigmented lesions especially in elderly people. The pathogenesis of drug-induced pigmentation is variable according to the causative medication and can involve an accumulation of melanin, sometimes following a nonspecific cutaneous inflammation and often worsened by sun exposure, an accumulation of the triggering drug itself, a synthesis of special pigments under the direct influence of the drug or deposits of iron following damage to the dermal vessels. The influence of sun exposure is usually obvious in most cases, either by sun-induced melanin synthesis stimulation with formation of complexes between melanin and the causative drug or by transformation of the drug in visible particles usually taken up by dermal macrophages under the influence of sunlight. The main drugs implicated in causing skin pigmentation are nonsteroidal anti-inflammatory drugs, antimalarials, amiodarone, cytotoxic drugs, tetracyclines, heavy metals and psychotropic drugs. Clinical features are very variable according to the triggering molecule, with a large range of patterns and shades which are sometimes more or less reminiscent of the culprit drug. Histological findings are very variable as well but the colored particles are often concentrated within dermal macrophages which are sometimes localized in a distinctive fashion with respect to dermal structures such as vessels or adnexes. Treatment is often limited to sun-avoidance or interruption of treatment with the offending drug but laser therapy recently gave rise to hope of a cure in some cases. These measures are often followed by a fading of the lesions but the pigmentation may last for a long time or may even become permanent in a small percentage of patients.

Adult↗
PubMed2005

Long-term efficacy and safety of tretinoin emollient cream 0.05% in the treatment of photodamaged facial skin: a two-year, randomized, placebo-controlled trial.

BACKGROUND: Long-term (>1 year) placebo-controlled studies of tretinoin in the treatment of photodamaged skin have not been conducted. Recently, we conducted a 2-year placebo-controlled study of tretinoin emollient cream 0.05%, including histopathologic assessment of safety and analysis of markers of collagen deposition. OBJECTIVE: The objective of the study was to determine the long-term safety and efficacy of tretinoin emollient cream 0.05% in the treatment of moderate to severe facial photodamage. METHODS: A total of 204 subjects were treated with tretinoin or placebo (vehicle emollient cream) applied to the entire face once a day for up to 2 years. Clinical and histologic effects were assessed at regularly scheduled clinic visits. RESULTS: Treatment with tretinoin resulted in significantly greater improvement relative to placebo in clinical signs of photodamage (fine and coarse wrinkling, mottled hyperpigmentation, lentigines, and sallowness), overall photodamage severity, and investigator's global assessment of clinical response (p<0.05). Histologic evaluation showed no increase in keratinocytic or melanocytic atypia, dermal elastosis, or untoward effects on stratum corneum following treatment with tretinoin compared with placebo. Immunohistochemistry studies, conducted at three study centers, showed a significant increase relative to placebo in facial procollagen 1C terminal, a marker for procollagen synthesis, at month 12 (p=0.0074). CONCLUSION: Long-term treatment with tretinoin emollient cream 0.05% is safe and effective in subjects with moderate to severe facial photodamage.

Administration, Topical↗
PubMed2004

Comparison of characteristics of acquired bilateral nevus of Ota-like macules and nevus of Ota according to therapeutic outcome.

Both acquired bilateral nevus of Ota-like macules (ABNOM) and nevus of Ota are characterized by the presence of dermal melanocytes. There are no differences in the method of treatment, however, postinflammatory hyperpigmentation (PIH) develops more often in ABNOM than in nevus of Ota following treatment. We investigated the differences in the development of PIH after treatment between ABNOM and nevus of Ota, and the histopathologic differences in the PIH. A total of 82 patients with ABNOM (n=47) and nevus of Ota (n=35) were treated with Q-switched alexandrite laser and followed up 2 weeks and 3 months later. Biopsies were performed on lesional skin before treatment. The distribution and the amount of melanin pigments were visualized with Fontana-Masson stain, and the distribution and the depth of melanocytes were measured by GP-100 (NK1-beteb) stain. Clinically, there was more erythema and PIH in ABNOM than in nevus of Ota. Histopathologically, intradermal melanocytes were clustered in groups and dispersed perivascularly in ABNOM, while melanocytes were scattered evenly throughout the dermis in nevus of Ota. Both groups show that when there is a statistically significant number of melanocytes in the perivascular area, erythema and PIH occur after laser therapy. In conclusion, indirect vessel injury in addition to perivascular clustering melanocytes might be considered the cause of increased PIH after treatment in ABNOM.

Adolescent↗
PubMedDate not supplied

Laugier-Hunziker pigmentation.

Laugier-Hunziker pigmentation (LHP) is an acquired disorder of hypermelanosis characterized by mucocutaneous hyperpigmentation. LHP may resemble various disorders characterized by mucocutaneous pigmentation. A 58-year-old lady presented with progressively increasing number of variable sized, hyperpigmented macules over the lips, fingers, toes and nails. There was no family history of similar illness. Systemic examination and all relevant investigations were within normal limits. Histopathology of a skin lesion had features consistent with LHP. The diagnosis of LHP must be made only after relevant investigations to rule out any associated systemic involvement. This case further highlights that LHP is not restricted to European countries.

Female↗
PubMed2004

The Laugier-Hunziker syndrome.

Laugier-Hunziker syndrome is a rare, acquired, benign hyperpigmentation of the lips, oral mucosa and nails. Although it is a benign disorder, other pigmentary disorders affecting the oral mucosa and nails must be considered in the different diagnosis. We presented a case of Laugier-Hunziker syndrome, showed clinical and histopathologic features of the disease.

Adult↗
PubMed1999

Skin pigmentary anomalies and mosaicism for an acentric marker chromosome originating from 3q.

We report on a 22 year old man with hyperpigmentation distributed along the lines of Blaschko in whom cytogenetic analysis showed mosaicism for an unusual supernumerary marker chromosome. The patient was of normal intelligence and was not dysmorphic. The marker was present in 30% of his lymphocytes and in 6% of his skin fibroblasts from a dark area, while fibroblasts from a light area showed a normal karyotype, 46,XY. We have identified the origin of the marker using fluorescence in situ hybridisation (FISH) with whole chromosome painting probes and YAC specific clones. The marker was found to consist of duplicated chromosome material from the distal part of chromosome 3q and was interpreted as inv dup(3)(qter-->q27.1::q27.1-->qter). Hence, this marker did not include any known centromeric region and no alpha satellite DNA could be detected at the site of the primary constriction. The patient was therefore tetrasomic for 3q27-q29 in the cells containing the marker chromosome. We postulate that, in our case, pigmentary anomalies may result directly from the gain of specific pigmentation genes localised on chromosome 3q.

Adult↗
PubMed2000

Erythema ab igne following heating/cooling blanket use in the intensive care unit.

Erythema ab igne caused by chronic heat exposure presents as a net-like hyperpigmentation of the skin. We report this condition rapidly evolving due to the inadvertent use or dysfunction of an adjustable-temperature blanket in the intensive care unit. The use of heating/cooling blankets, especially in a patient with altered mental status, can result in iatrogenic erythema ab igne.

Adult↗
PubMed2000

Linear atrophoderma of Moulin.

We report a typical case of linear atrophoderma of Moulin that represents a distinct clinical entity. A 17-year-old woman presented with hyperpigmented and atrophic band-like skin lesions measuring 3-5 cm in breadth on the right side of her trunk and on the right buttock, in an arrangement following the system of Blaschko's lines. The skin lesions had a normal texture and showed no signs of inflammation, lilac ring, erythema, induration, sclerosis or depigmentation. Routine laboratory data were normal. Antinuclear antibodies and anti-Scl70-antibodies were negative. Histopathologically, a moderate diffuse hyperpigmentation within the lower epidermis, a focal vacuolar degeneration of the basal layer, and a few dermal perivascular lymphocytes accompanied by signs of pigment incontinence were noted. Intravenous penicillin G was administered for 14 days in a dosage of 10 x 10(6) IU twice daily. This treatment was repeated after 3 and 9 months. No effect was noted. Linear atrophoderma of Moulin takes a chronic course without progression or regression. The disease leads to significant cosmetic impairment and this may cause emotional stress. Due to its relationship to idiopathic atrophoderma of Pierini-Pasini and linear scleroderma, treatment with intravenous penicillin was tried. In our case it had no effect.

Adolescent↗
PubMed2000

Cronkhite Canada syndrome.

Cronkhite Canada syndrome is an acquired non-familial syndrome characterised by diffuse gastrointestinal polyposis with alopecia nail dystrophy and hyperpigmentation. There is chronic diarrhoea and protein losing enteropathy. The etiology of this syndrome remains obscure. The rarity of the case prompts this case report.

Adrenal Cortex Hormones↗
PubMed2001

[Chronic arsenicism].

BACKGROUND: Arsenic is an ubiquitous natural element. Chronic and low level ingestion or inhalation may result in chronic arsenicism first characterized by skin changes. CASE REPORT: A 75 year old man, non-insulin-dependent diabetic, presented a diffuse hyperpigmentation with scattered white spots on the trunk. He complained of asthenia. Clinical diagnosis of chronic arsenicism was confirmed by arsenic determination in urine, plasma and phaneres. Thorough investigations led to discover very high arsenic levels in the own wine of the patient. This was probably the result of a wrong use of sodium arsenite-based fungicide, for cultivating his vine yard. DISCUSSION: Chronic arsenicism has become rare but it should always be kept in mind. Clinical presentation, with particular cutaneous features and routes of exposure are reviewed. Treatment is symptomatic. As arsenic is known to be a strong carcinogenic agent, patients with chronic arsenicism have to be followed up during a long time.

Aged↗
PubMed2001

Hemarthrosis as initial presentation of scurvy.

Vitamin C deficiency or scurvy is a disease now rarely seen except for certain populations at risk. When it occurs, diagnosis can be difficult as it can mimic other disorders. Its manifestations are primarily due to an abnormality in collagen formation causing bleeding in the skin, joints, muscles, or gastrointestinal tract and dystrophic hair deformities. We describe a case of scurvy in a 43-year-old man who presented with new onset hemarthrosis with no history of bleeding disorder. He was found to have perifollicular hyperpigmentation and corkscrew hairs, highly suggestive of scurvy. He admitted to completely eliminating fruits and vegetables from his diet and his serum vitamin C level was markedly decreased. Treatment with daily vitamin C supplement led to relief of symptoms and resolution of skin changes.

Adult↗
PubMed2001

Whipple's disease.

A 52 year old lady was admitted for progressive pedal oedema over a six year period and recent onset of hyperpigmentation. Laboratory investigations revealed that she was having a malabsorption syndrome with protein losing enteropathy. In view of associated arthralgia and higher mental function disturbances, a clinical diagnosis of Whipple's disease was postulated. Duodenal biopsy revealed infiltration of the lamina propria with PAS positive macrophages, suggestive of Whipple's disease. This case is being reported to highlight that Whipple's disease can manifest in the most unsuspecting manner and that early treatment can cure the patient.

Edema↗