Work-related toxic epidermal necrolysis?

A case of toxic epidermal necrolysis is described in an employee of a company that carries out plastic extrusion using various resins, including cellulose acetate, co-polyester, polyvinyl chloride, acrylonitrile-butadienestyrene, and polyethylene. Air sampling during normal operating conditions indicated only trace amounts of hydrogen chloride and the plasticizer diethylphthalate. However, pyrolysis products of resin plugs could include compounds such as formaldehyde, acrylonitrile, and chlorinated hydrocarbons that have been associated with previous case reports of either erythema multiforme, Stevens-Johnson syndrome, or toxic epidermal necrolysis. The development of toxic epidermal necrolysis in this worker was directly preceded by exposure to a vapor from a machine used to dry cellulose acetate. The problems in determining work relatedness and advising about return to work are discussed.

Primary cutaneous Langerhans cell histiocytosis in an adult.

An unusual adult case of Langerhans cell histiocytosis is presented. The case initially mimicked granuloma annulare, both clinically and histologically. The patient subsequently had development of extensive cutaneous involvement that pathologically revealed a diffuse infiltrate of CD1-positive histiocytic cells containing Langerhans granules. Extensive investigations failed to detect systemic involvement. The patient's cutaneous eruption did not respond to various therapeutic interventions, including phototherapy with oral psoralen with long-wave UV radiation in the A range (PUVA) and systemic vinblastine sulfate. Marked but temporary clinical and histologic improvement was achieved with total body electron beam radiotherapy. The nosology of this case is discussed in the context of the various histiocytic proliferative disorders.

Langerhans cell histiocytosis in monozygotic twins.

Langerhans cells histiocytosis, one of a group of histiocytosis syndromes characterized by Langerhans cell infiltration, has many clinical manifestations. In the past 30 years, numerous cases of presumed Letterer-Siwe disease, the acute multiorgan variant, have been reported in twins and siblings. Only recently has the Histiocyte Society established a criterion for a "definitive diagnosis" of Langerhans cell histiocytosis--the presence of Birbeck granules within the cells of the histiocytic infiltrate. We report the fatal outcome of Langerhans cell histiocytosis in monozygotic twin infants. There is no satisfactory explanation why Langerhans cell histiocytosis occurs concurrently in twins. We suggest that cytokines may provide an endogenous signal that triggers the pathologic proliferation of Langerhans cells.

Leprotic involvement of peripheral nerves in the absence of skin lesions. Case report and literature review.

In the absence of clinically apparent cutaneous lesions, primarily neural leprosy is uncommon. Primarily neural leprosy presents clinically as a peripheral neuropathy that most frequently affects motor nerves and that occasionally involves sensory nerves as well. The long incubation period for leprosy and its occurrence outside endemic areas often lead to delayed diagnosis. We present a case of glove and stocking hypoesthesia, weakness of the flexor muscle of the right great toe, palpable thickening of the right popliteal nerve, and hypoesthetic but normal-appearing areas on the back, which developed in a Trinidadian immigrant who lived in Canada for 16 years. A skin biopsy specimen obtained from a visibly normal but hypoesthetic area on the back demonstrated a few acid-fast bacteria in small dermal nerves, in arrector pili smooth muscle, and in rare perivascular histiocytes, associated with a sparse mixed inflammatory cell infiltrate. The patient responded well to therapy with dapsone, rifampin, and clofazamine. A classification and review of primarily neural leprosy is presented. Our patient represents the first reported case of primarily neural borderline lepromatous leprosy in Canada.

Pretibial epidermolysis bullosa: report of a case.

Pretibial epidermolysis bullosa is a rare variant of hereditary epidermolysis bullosa that is characterized by the delayed onset of a blistering eruption limited to the pretibial area. We present a case in a 48-year-old woman. Immunofluorescence mapping demonstrated blister formation below the basement membrane. This was confirmed by ultrastructural investigations that also revealed rudimentary anchoring fibrils similar to those described in the dominant dystrophic forms of epidermolysis bullosa.

Interleukin-1 enhances epidermal wound healing.

Previous in vitro studies suggest that the interleukin-1 (IL-1) proteins may be important for skin function. Keratinocytes are known to synthesize IL-1 in response to injury and IL-1 has been shown to stimulate fibroblast and keratinocyte growth, collagen synthesis by fibroblasts, and chemotaxis of keratinocytes. In order to evaluate the ability of IL-1 to enhance wound healing, recombinant human IL-1 alpha was applied topically in a water-miscible cream to partial thickness wounds made on the backs of pathogen-free adult pigs. The wounded site was excised at various times for assessment of epidermal regeneration. This study shows a statistically significant enhancement of healing of the IL-1-treated wounds compared with either air-exposed or vehicle-treated wounds. Histologic examination of biopsies from IL-1-treated wounds indicated complete and architecturally normal epidermal regeneration. Porcine keratinocytes and membranes prepared from porcine skin were found to possess high affinity receptors for human IL-1 alpha. This study suggests that topical administration of IL-1 may be useful for the promotion of wound healing.

Pseudoepitheliomatous, keratotic, micaceous balanitis. A clinical lesion with two histologic subsets: hyperplastic dystrophy and verrucous carcinoma.

A 58-year-old man presented with a 4-year history of a slowly enlarging, hyperkeratotic plaque on his glans penis that was compatible with a clinical diagnosis of pseudoepitheliomatous, keratotic, micaceous balanitis. The lesion had been treated unsuccessfully on three occasions with superficial shave excisional biopsies. Complete surgical excision produced excellent cosmetic and functional results with no evidence of recurrence at 3- and 6-month follow-up. A diagnosis of verrucous carcinoma was made on histologic examination of the excised material. Pseudoepitheliomatous, keratotic, micaceous balanitis is a distinctive clinical entity that represents a histologic spectrum ranging from hypertrophic-hyperplastic penile dystrophy to verrucous carcinoma.

Localized subepidermal bullae after intravenous phenobarbital.

The occurrence of subepidermal bullae and sweat gland necrosis in barbiturate-induced coma is well recognized. We report the case of a patient who received intravenous phenobarbital for refractory seizures and subsequently sustained subepidermal bullae without sweat gland necrosis in the skin around and proximal to the intravenous site. Although this may have been secondary to extravasation, a different mechanism of barbiturate-induced bulla formation may exist.

Allergic contact dermatitis due to urethane acrylate in ultraviolet cured inks.

Seven workers exposed to ultraviolet printing inks developed contact dermatitis. Six cases were allergic and one irritant. A urethane acrylate resin accounted for five cases of sensitisation, one of which was also sensitive to pentaerythritol triacrylate and another also to an epoxy acrylate resin. One instance of allergy to trimethylpropane triacrylate accounted for the sixth case of contact dermatitis in this group of workers. An irritant reaction is presumed to account for the dermatitis in the individual not proved to have cutaneous allergy by patch tests. In this instance trimethylpropane triacrylate was thought to be the most likely irritating agent. Laboratory investigation proved urethane acrylate to be an allergen. The results of investigations of the sensitisation potentials of urethane acrylate, methylmethacrylate, epoxy acrylate resins, toluene-2,4-diisocyanate, and other multifunctional acrylic monomers in the albino guinea pig are presented. The interpretation of such predictive tests is discussed.

Multiple cutaneous focal mucinoses with hypothyroidism.

A 54-year-old woman with myxedema had hundreds of discrete cutaneous mucinous papules, which responded dramatically to appropriate replacement therapy with L-thyroxine. Each lesion both clinically and histologically resembled a cutaneous focal mucinosis (cutaneous myxoma). Multiple cutaneous focal mucinoses are a previously undescribed manifestation of hypothyroidism that can be differentiated from other syndromes that have cutaneous mucinous deposits.