A double-blind study of twenty-eight patients with severe oral lichen planus treated with etretinate (75 mg daily) or a placebo for 2 months, showed that the oral retinoid had a marked beneficial effect. Nine non-responders who had received only placebo then entered an open cross-over study and they responded well to etretinate. Etretinate thus provided effective symptomatic relief for severe oral lichen planus, but side-effects were common, and six patients stopped treatment because of them.
Thirty two adult patients who presented with a sharply marginated, hyperkeratotic dermatitis of the palms (HDP), but with no skin lesions elsewhere, have been re-examined approximately 10 years later. Topical treatments had had limited effect. The morphological appearance and localization had remained unchanged, except in two individuals who were free from symptoms. One had developed psoriasis. The incidence of atopic disease and psoriasis in first degree relatives did not differ from that in control subjects. Mycological cultures and patch tests were negative. Serum IgE was normal. Skin biopsies in nine patients all revealed a chronic spongiotic dermatitis.
67 patients with oral lichen planus of the atrophic-erosive or reticular plaque type were examined. Dental amalgam in contact with mucosal lesions was present in 64 patients, and gold fillings in 33. Patch testing with a standard procedure was performed with components of dental fillings. 11 patients (16%) reacted to at least one of the mercury compounds compared to 8% in a reference group. Most positive reactions were caused by elemental mercury and ammoniated mercury. No patient reacted to gold or copper. Readings at days 10-14 did not increase the number of responders. 13 patients were patch tested with palladium; all were negative. It is not clear whether in the mercury-positive patients allergy to dental amalgam is a causative or aggravating factor, or merely on epiphenomenon.
Br. J. Ihrin. {\\)m) 81. r SUMMARY.-Autoerythrocyte sensitization syndrome (painful bruising syndrome) is a chronic purpuric cutaneous disease of women, characterized by localized painful inflammatory swellings which in a feAv hr. develop into painful ecchymoses. Some patients also have systemic manifestations: psychiatric and neurological symptoms, abdominal pain, and genito-urinary and gastrointestinal bleeding, in that order of prevalence.Histological examination shows an intense vaseulitis. Ecchymoses can occur spontaneously, particularly with emotional stress and after mild trauma. The haematological and blood clotting pictures are normal. Intradermal injection of erythrocytes or red cell stroma reproduces the spontaneous lesion.Two patients with this syndrome are presented. In both, typical ecchymotic reactions were induced by the injection of erythrocytes, in one a similar response could be provoked with phosphatldyl L-serine.The literature is reviewed in detail and the possible pathogenesis of the syndrome is discussed.IN 1955 Gardner and Diamond described in 4 women a s^Tidrome characterized by persistent, recurring, painful, ecchymoses which could be reproduced by intradermal injections of autologous erythrocyte suspensions. Since that time 27 additional cases have been reported, but because the syndrome lias attracted little attention in the European dermatological journals we are prompted to describe our 2 cases and to review the literature. CASE REPORTS Case 1An luuiiarriod woman, aged 27; she was an adopted child. In childlmod, slie occasionally had alxltjininal colic. She has always been nervous, aaxious and dependent. Menarche at 13. Menstruation wa.s at first regular, but, since 1960, has been irregular and sometimes profuse. In 1959, at 18, appendicectomy was performed. In the same year she left .school because of a " diflicult teacher ". At home tensioiis had arisen in lier relationship with her adopted mother. At this time she began to suffer from red, severely painful infiltrations on her arms and legs. The skin changes, within 24 hr., developed into large, painful bruises. On 3 occasions in 1960 she was admitted to the department of internal medicine. She had an impressive number of large, annular and circinate bruises, especially on tho arms and legs, hut also on the trunk, on the forehead and in one palm. Routine lal.)oratory investigations were normal. Treatment with antihistaminos and ACTH ivas Ai'ithout effect.Accepted for pubJieation Novemher 21st, 1968.
The syndrome of autoerythrocyte sensitization is characterized by painful ecchymotic lesions of the skin that develop in a very stereotypic manner. Onset occurs in a palm-sized skin region, usually on the limbs, with a stinging and burning sensation. After varying lengths of time, in general 2–6 hours, the skin becomes red, hot, oedematous and tender. This inflammatory phase may last anything up to twenty-four hours. Over the next few days the rubescent heated infiltration turns into an ecchymosis which spreads annularly at the same time as the tenderness and swelling diminish. Originally described by Gardner and Diamond (1955), thirty-one cases of the syndrome have now been reported. Up to the present all reported cases have come from North America and Australia and been confined to women with an age at onset of between 15 and 66 years.
Twenty-five patients with chronic oral lichen planus, usually of the atrophic-erosive type, were treated for 2 months with Etretinate® 0.6 mg/kg b.w./day, followed for 4 months by Etretinate® 0.3 mg/kg/day, or 0.1% Tretinoin® in an adhesive base. Complete resolution or improvement was seen in 85% of the lesions after first treatment. The number of patients with oral soreness and pain was significantly reduced (p< 0.001). During the second treatment, the improvement was maintained or even increased in about 70% of the patients, irrespective of the mode of treatment. Treatment was discontinued in one patient owing to moderately increased serum transaminase levels. The number of drug-related drop-outs was significantly lower than in a previous study utilizing an etretinate dose of about 1 mg/kg/day (p < 0.05). Other adverse effects were minor and tolerable. Retinoids offer an effective mode of therapy for severe oral lichen planus.
An unusual case of bullous pyoderma gangrenosum in a patient with myelodysplastic syndrome during treatment with granulocyte colony-stimulating factor (G-CSF) is reported. The possible relationship between G-CSF therapy and pyoderma gangrenosum, as well as the beneficial effect of cyclosporin A therapy, is discussed.
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