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Type III and type IV hypersensitivity reactions due to mitomycin C.

A 71-year-old man developed an exfoliative dermatitis of the palms of the hands and soles of the feet, and a generalized itch, during treatment with intravesical instillations of mitomycin C for an undifferentiated carcinoma of the bladder. Patch tests with mitomycin C 0.03%, 0.1% and 0.3% aq. were positive. Because of the serious consequences of this finding, the patient was retested with mitomycin C in pet. (same concentrations), a more stable preparation. This showed clear positive reactions. During this last series of patch tests, he developed palpable purpura on the legs. We postulated that this reaction was an immune-complex-mediated reaction, caused by the 2nd series of patch tests with mitomycin C. To prove this, we performed histopathological and immunofluorescence investigations, and these showed the reaction to be consistent with Henoch-Schonlein-type purpura. We therefore conclude that this patient developed systemic reactions to mitomycin C, characterized by an eczematous dermatitis as well as purpuric reactions. The intravesical installations with mitomycin C have been stopped. The patient's skin problems (the purpura as well as the eczema) have completely resolved and have not recurred.

Administration, Intravesical↗

[Distal cutaneous necrosis, an unusual etiology: hyperhomocysteinemia].

INTRODUCTION: Homocysteine is a sulfur amino acid occurring in methionine intermediary metabolism. It was recently shown to be a vascular risk factor even without high serum levels. CASE REPORT: A 29-year-old woman had painful plantar nodules and necrosis of the fifth toe on the right foot with cyanosis of the forefoot. The histology examination of a nodule biopsy gave the diagnosis of subcutaneous thrombophlebitis. Vascular explorations disclosed advanced-stage arteriopathy of the leg. The methionine loading test was abnormal and search for the heterozygous form of cystathionine ss was positive. DISCUSSION: Different diagnoses could be possible for cases associating an inflammatory venous disease and stage IV arteritis. Latent hyperhomocysteinemia was diagnosed in this case on the basis of the methionine loading test and identification of the heterozygous form of cystathionine ss. Patients with latent hyperhomocysteinemia have an increased risk of both thrombosis and arteritis. Our case emphasizes the importance of searching for hyperhomocysteinemia in patients with early arterial and/or venous thromboembolism, either as repeated episodes or occurring in an unusual territory.

Adult↗

Exophiala jeanselmei causing mycetoma pedis in India.

Exophiala jeanselmei causing mycetoma pedis in a 65-year-old Bengalee businessman was described. The right lower leg and foot were irregularly swollen, firm, painless, non-tender and had many small nodules and sinuses all over the swelling. The lower end of the tibia and bones of the foot were affected. The discharge from the sinuses and nodules contained black brown, soft, vermicular, crescent-shaped to irregular granules, measuring 0.5--2.00 mm and composed mostly of swollen spherical cells, 4-8 micrometer diameter, and a few hyphae, 2.5--3.0 micrometer diameter. The isolate was polymorphic in nature and exhibited various morphological features such as yeast-like cells producing secondary conidia, toruloid hyphae, and terminal and intercalary annellophores bearing annelloconidia at one or more points.

Aged↗

The treatment of progressive pigmented purpura with ascorbic acid and a bioflavonoid rutoside.

Progressive pigmented purpura (PPP) is a group of dermatoses that are benign and usually self-limited. However, they may persist for months or years with frequent recurrences. Numerous treatments have been tried, but no effective therapy has yet proven to be successful. This report documents the treatment of a patient with one subtype of PPP using ascorbic acid and a bioflavonoid rutoside given orally. A 42-year-old male with biopsy-proven Eczematoid-like Purpura of Doucas and Kapetanakis was treated with ascorbic acid 500 mg twice a day and a bioflavonoid rutoside 50 mg twice a day. Treatment was successful, with quick defervescence of the rash and no recurrence to date.

Administration, Oral↗

[Idiopathic white atrophy causing cutaneous thrombotic microangiopathy. Apropos of a case].

Authors report clinical data on a 59-year-old woman, bearing, since 15 years, painful purpuric and necrotizing macules and papules on the legs, leading to white atrophic scars. On examination one could see lesions in various stages of development. Clinical examination and all laboratory investigations were otherwise normal. First cutaneous biopsy led to an erroneous diagnosis of vasculitis with subsequent corticotherapy. Second biopsy displayed numerous fibrinous thrombi inside lumens of superficial dermal capillaries without fibrinoid necrosis of their parietal layer nor inflammatory infiltrate. A diagnosis of white atrophy was made on the coexistence of clinical and histopathological data, according to Shornick's criteria. Drastic improvement of clinical condition was obtained with heparin therapy. White atrophy is a clinicopathological entity first described in 1929 by Milian, then by Nelson in 1955. Authors describe here its main clinical data with diagnostic considerations and pathophysiology.

Atrophy↗

Eczema herpeticum during treatment of atopic dermatitis with 0.1% tacrolimus ointment.

UNLABELLED: Recent evidence suggests that 0.1% tacrolimus ointment is an effective treatment of atopic dermatitis. Tacrolimus is an immunosuppressive agent that interferes with cell-mediated immunity. We have observed 2 cases of eczema herpeticum among 36 patients with atopic dermatitis treated with a topical preparation containing 0.1% tacrolimus. A 29-year-old male patient developed generalized herpetic lesions on his face on the 4th day of treatment. His SCORAD was then 73, and the tacrolimus blood level was 7.5 ng/ml. A 23-year-old woman developed disseminated herpetic lesions on her neck, face, shoulders and legs during the 9th week of treatment. Her SCORAD was then 41, tacrolimus blood levels were <3 ng/ml 2 weeks before the infection. Herpes simplex virus type 1 antigens were identified in several lesions by direct immunofluorescence in both patients. Neither patient recalled previous episodes of cold sores. The lesions resolved quickly under intravenous acyclovir treatment but resulted in important facial scarring in the male patient. CONCLUSIONS: Eczema herpeticum is a well-known complication of atopic dermatitis. Available data do not allow to link topical tacrolimus with an increased risk for eczema herpeticum, but they are insufficient to exclude an association. Future studies and careful documentation of cases are needed in order to better characterize patients at risk.

Adult↗

Giant verruca vulgaris.

BACKGROUND: Common warts (verrucae vulgares) are hyperkeratotic papulonodules that are most often seen on the hands, arms, and legs, but they can be seen anywhere on the glabrous skin. Common warts represent the most frequent clinical lesions produced by the human papillomavirus (HPV). OBJECTIVE: Although they are diagnosed easily, giant, chronic, isolated, solitary, recalcitrant verruca vulgaris involving nail and nail bed on the digits can be misdiagnosed, and the differential diagnosis should be made carefully and verified by biopsies. METHOD: A 50-year-old male patient had a giant periungual verruca vulgaris on his left second finger. It was present for 2 years, was unresponsive to any treatment modality, and was treated by surgical excision. RESULTS: After 18 months of follow-up, there was no evidence of recurrence, and both aesthetic and functional results were quite satisfactory. CONCLUSION: Lesions of long duration generally lose their characteristic histopathologic features, and HPV cannot be detected in these lesions by polymerase chain reaction. Therefore, multiple sections should be examined histopathologically, and extreme care should be taken in order to avoid misdiagnosis.

Fingers↗

Lichen planopilaris.

P, a 20-year-old laborer displayed initial symptoms of the disease in question when he was 10 years old. Initially he had an asymptomatic progressive loss of hair on the scalp. A couple of years later he had mild to moderate pruritus, and the appearance of slate-blue eruptions on the scalp and elsewhere on the body. This resulted in a complete loss of hair on the vault of the scalp, which led him to seek specialist opinion. Skin surface examination revealed the presence of grayish-blue acuminate follicular papules, disposed singly and in groups (plaques). The pilo-sebaceous orifices were conspicuously obliterated and filled by keratin plugs. Perifollicular erythema was a predominant feature on the scalp. The lesions were present over the scalp, around the neck, chest, back, axillae, groin and legs. Shiny atrophied scalp skin depicting scarring alopecia mimicking male-type baldness was a salient feature. In addition, it was studded with conspicuous acuminate papules in its center (Fig. 1a). The known nonhairy (glabrous) skin had classic lichen planus lesions (Fig. 1b). Hemotoxylin-eosin stained microsections prepared from typical lichen planus (LP) lesions over the abdomen and those of lichen planopilaris (LPP) of the scalp were simultaneously studied. The former revealed changes in the epidermis comprising of hyperkeratosis, increase in thickness of stratum granulosum, hydropic degeneration of the basal cell layer and band-like lympho-histiocytic infiltrate pressing against and invading the epidermis, while the latter revealed uniform atrophy of the epidermis and vacuolization of basal cells. The hair follicles were dilated and were filled with keratin plugs. In addition to fibrosis of the dermis, pigment laden microphages and lympho-histiocytic infiltrate was prominent. The follicles and the sebaceous glands were absent. However, arrectores pilorum and sweat glands were preserved (Fig. 2a,b).

Adult↗

Branched filaments no fungus, ovoid bodies no bacteria: Two unusual cases of mycetoma.

We describe a 58-year-old man presenting with necrotizing panniculitis of the lower right leg and a 64-year-old woman with a clinically similar lesion combined with pustular eruptions and subsequent ulceration on the forehead. In the first patient, Giemsa staining showed small ovoid bodies and Grocott staining revealed hyphae. Histology from the process on the forehead showed branched filaments in the periodic acid-Schiff (PAS) staining. In the first case, Madurella mycetomatis, a fungus, was the pathogenic agent, whereas in the other case white colonies of filamentous organisms resembling fungi could be cultivated that turned out to be the bacterium Nocardia brasiliensis. Since the initial clinical appearance of these two forms of mycetoma were almost identical and histopathologic findings were inconclusive, only sophisticated microbiologic work-up of material from lesional skin led to the correct diagnosis. In times of global tourism, these unusual cases impressively document the necessity to become more familiar with mycetoma to make accurate therapeutic decisions with effective results, possibly saving a limb.

Antifungal Agents↗

A case of mixed bullous disease of epidermolysis bullosa acquisita and linear IgA bullous dermatosis.

A 75-year-old Japanese male visited us with bullous eruptions on the extremities. Physical examination revealed large bullae on the hands, lower legs and feet. The oral mucosa was also involved. Histology disclosed subepidermal blister with inflammatory cell infiltrates in the dermis. Direct immunofluorescence showed deposits of IgG and IgA at the cutaneous basement membrane zone. Indirect immunofluorescence on 1 M NaCl-split human skin sections demonstrated that the patient's IgG antibodies reacted with the dermal side of the split, while IgA antibodies reacted with the epidermal side. Immunoblotting showed that the patient's serum reacted with the NC1 domain of type VII collagen (290-kDa epidermolysis bullosa acquisita antigen) as well as the 120-kDa linear IgA bullous dermatosis antigen, LAD-1. Systemic prednisolone resulted in a favorable response. From the clinicopathological findings, the present case is not consistent with either epidermolysis bullosa acquisita or IgA bullous dermatosis. Therefore, we regarded the case as mixed bullous disease of epidermolysis bullosa acquisita and linear IgA bullous dermatosis. Such a case has not been previously reported.

Administration, Oral↗

Increased levels of inflammatory cytokines in human skin lymph derived from sodium lauryl sulphate-induced contact dermatitis.

A superficial peripheral lymph vessel draining the skin of the upper and medial part of the foot was cannulated on the lower leg of six healthy human volunteers. After 2 days an irritant contact dermatitis was induced by application of 10% sodium lauryl sulphate to the area of skin drained by the lymph vessel. Three days later the spontaneously regressing skin reaction was treated with clobetasol propionate. Lymph was collected twice daily for 7 days, and the levels of various cytokines (IL-1 alpha, IL-1 beta, IL-2 and soluble IL-2 receptors, IL-6, IL-8, TNF-alpha, GM-CSF) were determined by ELISA technique. In the majority of the volunteers all cytokines examined were detected in several lymph samples, with the exception of IL-1 alpha and IL-8. In parallel with the clinical symptoms of the contact dermatitis the levels of IL-6 and TNF-alpha increased 8-10-fold, whereas for IL-1 beta, IL-2, IL-2 receptors, and GM-CSF there was a delayed, 2-3-fold increase. These results suggest that cytokines, in particular IL-6 and TNF-alpha, may actively participate in the immunological reactions in the skin and in the regional lymph nodes during contact dermatitis.

Cytokines↗

Interdigital lesions and frequency of acute dermatolymphangioadenitis in lymphoedema in a filariasis-endemic area.

BACKGROUND: Lymphatic filariasis (LF) is a mosquito-borne nematode infection that causes permanent lymphatic dysfunction in virtually all infected individuals and clinical disease in a subset of these. One major sequel of infection is lymphoedema of the limbs. Lymphoedema of the leg affects an estimated 15 million persons in LF-endemic areas worldwide. Acute dermatolymphangioadenitis (ADLA) in people with filarial lymphoedema causes acute morbidity and increasingly severe lymphoedema. Episodes of ADLA are believed to be caused by bacteria, and it has been shown that entry lesions in the skin play a causative role. Clinical observations suggest that interdigital skin lesions of the feet, often assumed to be fungal, may be of particular importance. OBJECTIVES: To investigate the epidemiology and aetiology of interdigital lesions (IDL) of the feet in filarial lymphoedema. METHODS: The frequency and mycological aetiology of IDL in 73 patients with filarial lymphoedema were compared with 74 individuals without lymphoedema in a region of Guyana highly endemic for Wuchereria bancrofti. RESULTS: More than 50% of patients with lymphoedema had one or more IDL (odds ratio 2.69; 95% confidence interval 1.31-5.66; P<0.005 compared with controls). The number of lesions was the strongest predictor of frequency of ADLA. Only 18% of the lesions had positive microscopy or culture for fungi (dermatophytes and Scytalidium). CONCLUSIONS: These findings highlight the importance of interdigital entry lesions as risk factors for episodes of ADLA and have implications for the control of morbidity from filarial lymphoedema.

Acute Disease↗

Using silicone gel sheet for the treatment of facial telangiectasias with sclerotherapy.

BACKGROUND: Telangiectasias are superficial cutaneous vessels that are visible to human eye and are present as a dilated venule, capillary, or arteriole. They are a common cause of concern, especially when located in face. Sclerotherapy is among the treatment alternatives, but facial telangiectasias are less responsive than those located on the leg. OBJECTIVE: To show the effectiveness of silicone gel sheet in improving the results obtained by sclerotherapy in facial telangiectasias. METHODS: Between 1998 and 2003, 32 patients were treated with sclerotherapy and silicone gel sheet (group 1). Twenty-six patients were treated with sclerotherapy and cotton ball (group 2), and 30 patients were treated only sclerotherapy (group 3). Patients were evaluated for 12 weeks for the degree of improvement and side effects. All complications, side effects, and number of sessions were also recorded at each follow-up visit. The number of sessions is limited to six. After the final follow-up visit, the photographs taken of each treatment site at baseline and at final visit were reviewed in a double-blinded manner based on a 0 to 4 scale by two physicians. RESULTS: Fifty one of the 88 patients showed improvement totally. In the silicone sheet-sclerotherapy group, 22 of the 36 patients showed improvement. In the sclerotherapy-cotton ball group, 14 of the 26 patients showed improvement, and in the sclerotherapy-only group, 15 of the 30 patients showed improvement completely within the 12 weeks. The patients treated with sclerotherapy-silicone gel sheet (group 1) had more improvement than the other groups. CONCLUSION: Combining silicone gel sheeting to sclerotherapy increases the success rate in treating facial telangiectasias, especially facial subunits, which have bone support.

Adolescent↗

Treatment of face veins with a cryogen spray variable pulse width 1064 nm Nd:YAG Laser: a prospective study of 17 patients.

BACKGROUND: Through the 1990s laser treatment of leg veins has been a challenge. Newer, longer wavelength lasers with variable pulse width have shown promising results for both telangiectasia and reticular leg veins. Experience with these lasers for treatment of facial telangiectasia and periorbital reticular veins is scant. OBJECTIVE: To our knowledge this is the first prospective study to evaluate a variable pulse width, cryogen spray-equipped 1064 nm Nd:YAG laser for the treatment of facial telangiectasia and larger periorbital reticular veins. METHODS: Seventeen volunteers, ages 32-67 years, skin types I-IV, with facial telangiectasia and reticular periorbital/temporal veins were treated once with the Nd:YAG laser at fluences of 125-150 J/cm2 with a 6 mm spot size and pulse widths of 25 msec for small vessels and 75-100 msec for reticular veins. Patients were examined 1 week and 1 month after the treatment. Pre- and posttreatment 35mm photographs were taken. Improvement was judged by two experienced physicians, both visually on patients and by comparison of pre- and posttreatment photographs. Results were graded as percent resolution, in five groups: 0%, 0-25%, 25-50%, 50-75%, and 75-100%. RESULTS: All 17 patients completed the study. Thirty-two sites were treated (24 cheek, nose, and chin telangiectases, and 8 periorbital reticular veins). Greater than 75% improvement was observed in 97% of the treated sites. Greater than 50% improvement was observed in 100% of the treated sites. Notably, 100% of the facial reticular veins treated have essentially 100% resolution. Pain during treatment was variably perceived by patients, but occasionally sufficient for patients to express reservations about additional future treatment. Transient erythema and edema were common, but fine crusting was rare. Small areas of purpura were also quite rare. CONCLUSION: The cryogen spray-equipped 1064 nm Nd:YAG laser was remarkably effective and safe for skin type I-IV patients. It is an excellent tool for treatment of facial telangiectasia with minimal risk of purpura. For the first time we appear to have a simple, safe, and effective treatment for 1-2 mm periorbital reticular veins. The use of topical anesthesia may be needed for some patients.

Adult↗