Biomedical subjects
Publications and source records attributed to D J Gawkrodger.
Four patients with white sponge naevus of the oral mucosa each showed a dramatic improvement in symptoms with the use of topical tetracycline. This effect may depend on antimicrobial activity but the exact mechanism remains uncertain.
Syringoacanthoma is a rare benign appendage tumour derived from the intraepidermal portion of the acrosyringium. We report its occurrence in 3 women. In each case the tumour clinically most resembled a seborrhoeic keratosis. Its importance histopathologically lies in the need to differentiate it from conditions such as malignant melanoma, squamous cell carcinoma, intraepidermal carcinoma, Paget's disease and metastasis, which may all show intraepidermal spread.
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Two patients with lichen planus pemphigoides and two with bullous lichen planus were compared. Lichen planus pemphigoides was clinically distinguished by a more generalized lichen planus, more extensive blistering, the need for systemic corticosteroids and by a longer course. The blister of bullous lichen planus was a subepidermal bulla showing degeneration of the epidermal basal layer and other features of lichen planus, whereas in lichen planus pemphigoides the bulla was similar to that of bullous pemphigoid albeit with rather more neutrophils than are usually seen. Direct immunofluorescence was positive in lichen planus pemphigoides and negative in bullous lichen planus. Lichen planus pemphigoides and bullous lichen planus are separate entities: the former is an auto-immune disease precipitated by lichen planus and not related to bullous pemphigoid, the latter is probably not auto-immune but represents the extreme consequence of the lymphoid infiltrate at the dermo-epidermal junction.
Six hours after challenge with dinitrochlorobenzene (DNCB) in sensitized volunteers, infrequent cells reacting with both OKT6 (CD 1a) and with an antibody to DNCB were demonstrated in the upper dermis in 3 out of 4 subjects, using a double fluorescence technique. Single marker studies using polyclonal and monoclonal antibodies to DNCB showed cytoplasmic reactivity with peripheral accentuation in keratinocytes throughout the epidermis, most intense in the granular and basal layers, and occasional positive cells in the papillary dermis. Immunoperoxidase electron microscopy using polyclonal and monoclonal antibodies to DNCB demonstrated reactivity on keratinocyte and Langerhans cell membranes, with intracellular deposition of reaction product on mitochondria. Reaction product was present on the external leaflets of Birbeck granules, suggesting that the penetration of DNCB was passive rather than due to an active endocytic process. The immunofluorescent studies suggest that the double-fluorescing upper dermal cells were Langerhans cells but this could not be confirmed by electron microscopy.
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Nutritional status of 86 patients with dermatitis herpetiformis (DH) was defined by anthropometric measurements and hematological and biochemical laboratory tests to establish prevalence of malabsorption and malnutrition. Anthropometric measurements in DH patients were comparable to normal control patients. Four individuals were of short stature; two had had diarrhea and failed to thrive in childhood. Abnormalities attributable to nutritional deficiency were detected in only 6 of the 86, whereas drug-associated hematological or biochemical changes were present in 36 of 55 subjects treated with dapsone or sulfapyridine. Twenty patients had hemolytic anemia or macrocytosis related to drug therapy. Only two had anemias attributable to malabsorption; one was iron deficient, the other folate deficient. Two other patients were mildly Fe deficient and two had slight folate deficiency; they lacked other stigmata of malabsorption. Drug-induced hematological and biochemical abnormalities were more common than changes that suggest nutritional disease, even though most DH patients had an enteropathy at presentation.
Plasma 5-S-cysteinyldopa (5-S-CD) concentration measured in healthy volunteers in Edinburgh, Scotland (latitude 56 degrees N) showed only minor changes during the day. However, when measurements were performed over a 12-month period a significant rise in 5-S-CD concentration was found. Skin pigmentation and hair colour were not related to plasma 5-S-CD levels. Patients with psoriasis treated with ultraviolet-B or photochemotherapy (PUVA) developed an almost two fold increase in their plasma 5-S-CD level within the first five treatments, before pigmentation developed, subsequent increments of up to four times the pretreatment level being found in the PUVA group. Dithranol treatment caused an increase in plasma 5-S-CD in some psoriatic patients, suggesting a possible association between skin erythema and elevated 5-S-CD levels. The value of plasma 5-S-CD in the follow-up of patients with malignant melanoma does not seem to be invalidated by unavoidable exposure of the subjects to sunlight in a temperate climate such as that of South East Scotland.
We report details of five patients with nail dystrophy associated with sarcoidosis. All had bone cysts in the underlying terminal phalanx and had a chronic course of the disease. Radiological examination is indicated in patients with sarcoidosis and nail dystrophy as bone cysts are frequently asymptomatic.
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Fifty patients with bullous pemphigoid were investigated over periods of up to 7 years. Sequential studies of circulating basement membrane zone (BMZ) antibody titres revealed two groups of patients: 44% had a high initial titre (greater than 1:160) and 56% had a low initial titre (less than 1:160). Subjects with titres greater than 1:160 continued to have high titres, whereas in those with lower titres the BMZ antibodies usually became undetectable after 4 months. The BMZ antibody titres were of no prognostic value. High titres remained high even in patients who were in remission and off treatment, and in these subjects, there was no evidence of a change in the IgG subclass. The initial BMZ IgG antibody titres showed a highly significant correlation with total serum IgE concentrations. A significant correlation also existed between total serum IgE and IgG4 concentrations, although not between the serum IgE and the peripheral blood eosinophil count. No anti-BMZ antibody of IgE class was found, nor specific IgE against inhalants and foods identified, and the significance of the high total serum IgE remains unexplained. The majority of those followed for more than 2 years were able to stop their steroid therapy without further blistering: this confirms that pemphigoid tends to be a self-limiting disease. Nine patients had a recent history of malignant disease, and this is no more than would be expected for this age group of patients: however, 3 patients presented with pemphigoid and figurate erythema and all died of neoplastic disease, suggesting that such patients should be investigated thoroughly for neoplasia.
212 epitheliomata developed in two unrelated women with self-healing epitheliomata of Ferguson-Smith. Most of the lesions appear on the face, often in crops; they enlarge over 4 weeks and, without treatment, resolve over 4 months to leave a ragged scar. One of the patients developed an unusual syringoma, a papillary carcinoma of the thyroid and a mucosal squamous cell carcinoma which failed to involute spontaneously. Cytogenetic studies did not reveal any abnormality of DNA repair. Surgical excision of the epitheliomata is successful but time consuming. X-irradiation should not be used, but cryotherapy of early lesions is helpful. One patient, treated with etretinate for 24 months, has developed only 8 epitheliomata over this time, compared with 23 in the previous 24 months. Etretinate therefore appears to prevent new lesions from developing.
A 27-year-old nickel-sensitive female who had had continuous spontaneous flare-ups of eczema, including at sites of previous metal contact, experienced a clearing of her eruption after commencing a low-nickel diet. When on the diet, whole-blood and urinary nickel levels fell to half or less of pre-diet values and this coincided with the clinical improvement. Low-nickel diets should be considered for patients who are highly nickel sensitive.
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