Erythema multiforme associated with cytomegalovirus infection in nonimmunosuppressed patients.

BACKGROUND: It is widely known that cytomegalovirus (CMV) primarily brings about subclinical and asymptomatic infection in the early stages of life and can cause various dermatological and systemic disorders under immunosuppressed conditions. Nonimmunosuppressed individuals very rarely present with cutaneous CMV involvement. OBJECTIVE: In the present study, we described the clinical characteristics of 5 nonimmunosuppressed adult patients with positive IgM antibody to CMV. METHODS: The systemic symptoms and dermatological features of these 5 patients were described. Laboratory examinations including blood cell counts, liver and renal functions were performed. IgG and IgM antibodies to CMV were also examined at the first consultation and 2-3 months after the skin eruption. Polymerase chain reaction for CMV DNA was performed in the skin samples of the patients. RESULTS: All 5 patients had fever and complained of a sore throat. Multiple exudative erythema and target lesions with itching were observed mainly on the extremities. These symptoms and eruptions disappeared within 1 week after the onset and IgM antibody titers significantly decreased after 2-3 months. IgG antibody to CMV was already positive in 3 cases but was negative in 2 cases at the initial consultation. CONCLUSION: We propose that CMV infection may cause erythema multiforme by primary, recurrent infections or reactivation of CMV even in nonimmunosuppressed adults.

Skin metastasis of breast cancer clinically undistinguished from amyopathic dermatomyositis.

We report a 65-year-old woman who consulted us on May 25, 1998, showing pruritic, partially flagellate erythema on the back and upper extremities, livedo lesions with erythema on the loins, and erythematous papules on the dorsal finger joints for 2 months. Histopathological findings of erythema on the back showed mononuclear cell infiltration around capillaries and marked edema in the dermis. Laboratory data were within normal range except for positive anti-nuclear antibody. She had undergone total left mastectomy on June 2, 1997 for breast cancer. Supraclavicular lymph node metastasis was found at the beginning of May, 1998. A diagnosis of amyopathic dermatomyositis associated with breast cancer was made. Erythema with itching gradually subsided from the end of August, 1998. Treatment with radiation and chemotherapy reduced lymph node swelling, but complete remission was not obtained. Erythema similar to the previous lesion but without itching re-appeared on the back from January, 2000. Histological findings of erythema showed many carcinoma cells similar to the primary lesion of left breast cancer in the whole dermis. A diagnosis of skin metastasis of breast cancer was made. These findings suggest that skin metastasis should be taken into account for patients with erythema on the trunk similar to dermatomyositis.

Pemphigus of the eyelids.

We report the case of a 56-year-old woman who presented with a 2-month history of widespread oral erosion and a 3-day history of small papules on the lower eyelids. No other skin involvement was found. Histopathological examination revealed suprabasal cleft and acantholysis in the lower epidermis of the papule on the lower eyelid and in the lower mucous membrane of the oral mucosa. Intercellular deposits of IgG and C3 were seen in the whole epidermis of the specimen from the papule on the right lower eyelid by direct immunofluorescence study. These deposits were also observed in the biopsy specimen from erosion on the left buccal membrane. Indirect immunofluorescence study using normal human skin as a substrate showed intercellular antibodies directed to the cell surface of the whole epidermis with a titer of 1:40. The titers of antibodies to desmoglein 3 and 1 were 118 and 25.9, respectively, by enzyme-linked immunosorbent assay. The patient was treated with an oral administration of prednisolone (0.75 mg/kg/day) for 9 days, which improved the skin eruptions and oral erosion. The dose of prednisolone was gradually tapered and it took 10 weeks to cease this treatment. These findings suggest that this patient is an unusual case of pemphigus vulgaris (mucosal dominant type) diagnosed from the clinical and histopathological findings, with positive antibodies to desmoglein 3 and 1.

Mixed connective tissue disease following interstitial cystitis.

A 64-year-old woman complained of severe infrapubic pain and pollakisuria with nausea, vomiting and diarrhea, but with normal urinalysis since 1987. The clinical diagnosis of interstitial cystitis (IC) was made, and cystectomy was performed in 1996. The bladder taken was markedly shrunken with a capacity of 50 ml, and showed bleeding on the mucosal surface. Histological findings of the bladder showed ulcer formation in the mucous membrane, and marked infiltration of mononuclear cells, edema and fibrosis in the submucosal tissue. She had noticed exudative erythema, swelling and sclerosis on the bilateral fingers and dorsal aspects of the hands since 1993, and Raynaud's phenomenon and morning stiffness of the fingers from November, 1998. Laboratory data showed positive anti-nuclear antibody (titer: 1: 2,560) and anti-U1 RNP antibody (titer: 69.5 by ELISA). A diagnosis of mixed connective tissue disease following IC was made.

"Apocrine" poroma: review of the literature and case report.

BACKGROUND: "Apocrine" poroma has recently been proposed as a new term to designate a distinctive benign skin neoplasm with differentiation toward the folliculosebaceous-apocrine unit. CASE REPORT: In support of alternative differentiation, a case of apocrine poroma is reported in a 73-year-old man. A nodule on the right upper abdomen, which was thought clinically to be seborrheic keratosis or basal cell epithelioma, was excised. HISTOLOGY: Histologically, this neoplasm heterogeniously consisted of poroma-like, sebaceous and follicular epithelial components. Since these components share the common embryologic origin of the folliculosebaceous-apocrine unit, this histologic pattern indicates apocrine differentiation of the tumor. CONCLUSION: This is an additional case to support the term "apocrine" poroma as a diagnosis.

Squamous cell carcinoma arising from lesions of porokeratosis palmaris et plantaris disseminata.

We report a 63-year-old Japanese man with numerous hyperkeratotic papules of porokeratosis palmaris et plantaris disseminata (PPPD) who developed multiple squamous cell carcinomas on the lesional sites of the palms and soles. The hyperkeratotic papules, which showed tightly packed columns of parakeratotic cells in the cornified layer (cornoid lamella), lost granular layer, and dyskeratotic keratinocytes in the epidermis below the cornoid lamella histologically, had been noticed on the palms and soles from the age of 28 and 43, respectively. He has no family history of such hyperkeratotic papules. Treatment with etretinate (10-50 mg/day) was given discontinuously, and the total dose of etretinate amounted to approximately 21 g over 14 years (average: 0.07 mg/kg/day). He noticed erosions on the hyperkeratotic papules on the left sole and palm more than 9 months after cessation of treatment with etretinate. Histological findings showed numerous atypical keratinocytes in the epidermis and upper dermis with mononuclear cell infiltration seen in the upper dermis. The diagnosis of squamous cell carcinoma arising from the lesions of porokeratosis palmaris et plantaris was made. Five erosions with histologically malignant changes were removed 1 cm from the margin of the erosions. These findings suggest that etretinate may have an inhibitory action on malignant changes in PPPD.

Tick bite by Haemaphysalis megaspinosa - first case.

We describe the first case of tick bite by Haemaphysalis megaspinosa. The tick was found on the skin at the right occipital area in a 5-year-old girl, who had gone on a picnic twice to a mountain 1 and 5 days before she noticed the tick. The tick was surgically removed. The tick was identified as an adult female of Haemaphysalis megaspinosa from its morphological characteristics. This is the first report of tick bite by Haemaphysalis megaspinosa.

Prevalence of normal-tension glaucoma and primary open-angle glaucoma in patients with collagen diseases.

PURPOSE: To investigate the prevalence of normal-tension glaucoma (NTG) and primary open-angle glaucoma (POAG) in patients with collagen diseases and determine whether an immunocompromised condition is present in a subset of glaucoma patients. METHODS: Three glaucoma specialists prospectively examined patients with collagen diseases. The diagnostic process included applanation tonometry, slit-lamp examination, gonioscopy, direct ophthalmoscopy, and automated static perimetry. Twenty-four-hour intraocular pressure monitoring was done when necessary. Using the results of a population-based survey conducted in Japan, we calculated an expected number of cases of NTG and POAG, and compared these with the actual number of cases. RESULTS: Of the 153 patients with collagen diseases examined, we found 6 patients with NTG and 2 patients with POAG. Of these 8 patients, 2 with progressive systemic sclerosis (PSS), one with NTG, and the other, POAG, had a history of being on systemic steroidal therapy. The prevalence of NTG and POAG was significantly higher in women patients having collagen diseases as compared with normal women (P = .027). CONCLUSION: Women patients with collagen diseases are highly susceptible to NTG and POAG.

The effect of electrical acupuncture-stimulation therapy using thermography and plasma endothelin (ET-1) levels in patients with progressive systemic sclerosis (PSS).

In all 11 patients with progressive systemic sclerosis (PSS; Barnett type I, 2; type II, 5; type III, 4 cases; male 1; female 10 cases; 45.2 +/- 10.2 years-old), 6 cases of scleroderma spectrum disorders (SSD, male 1; female 5 cases; 51.2 +/- 13.2 years-old) and 7 healthy controls (HC, male 1; female 6 cases; 43.1 +/- 8.4 years-old) were entered to be examined. The plasma endothelin-1 (ET-1) levels of PSS, SSD and HC were 1.98 +/- 0.69, 1.76 +/- 0.39 and 1.15 +/- 0.38 pg/ml, respectively. After the stimulation with the low frequency electrical current, electrical acupuncture, for unilateral side of hand/arm (30 min), the plasma ET-1 levels decreased in 10 cases of PSS treated (1.61 +/- 0.45 pg/ml), but no change of plasma serotonin levels. In 4 of 6 cases of SSD, plasma ET-1 levels increased (2.06 +/- 0.39 pg/ml), however, nitrate levels increased and serotonin decreased in 3 of 5 cases of SSD. In 6 cases of HC treated with the electrical acupuncture, the plasma ET-1 levels increased (1.72 +/- 0.58 pg/ml). Thermographically, 9 of 11 cases of PSS and 5 of 6 cases of SSD showed temporally temperature-elevation of hand/fingers not only in treated sides, but also in non treated sides, although none of 7 HC showed temperature-elevation of hands/fingers. The decrease in plasma ET-1 levels due to the electrical acupuncture was thought to induce the vasodilatation and elevate the surface temperature in patients with PSS. These results will provide an excellent basis to study the efficacy of electrical acupunctural stimulation.