Parry-Romberg syndrome is an extremely rare connective tissue disorder. It might be a form of linear scleroderma, and it manifests as progressive hemifacial atrophy, epilepsy, exophthalmos or alopecia. Herein we report a case of Parry-Romberg syndrome. A 68-year-old woman had left hemifacial atrophy, shortening and deformity of the left leg, and deformities to her side fingers that occurred from her 1st decade to 2nd decade. Sclerotic change had stopped spontaneously when she was 20 years old. Histopathologically, there were a few signs of skin appendages, but no sclerotic change. On autoimmune antibody test, no positivity was shown. X-ray showed shrinkage of the lung field, elevation of the left diaphragm, shortening of length and reduction of bone mass in the left femur, tibia, and fibula. And there were multiple melorheostasis. Neck CT showed left facial sclerotic change, bone mass loss, and multiple melorheostasis. Because the progression of sclerosis had stopped the patient did not receive any treatment.
Aged ; Alopecia ; Congenital Abnormalities ; Connective Tissue ; Diaphragm ; Epilepsy ; Exophthalmos ; Facial Hemiatrophy ; Female ; Femur ; Fibula ; Fingers ; Humans ; Leg ; Lower Extremity ; Lung ; Neck ; Scleroderma, Localized ; Sclerosis ; Skin ; Tibia

BACKGROUND: Exposure to Rhus (Japanese lacquer) can cause many adverse effects such as contact dermatitis, pruritus, rash and even hepatitis in human, and these effects can be caused by contact or ingestion of Japanese lacquer. Many Koreans currently eat Japanese lacquer boiled with chicken although it has many side effects. Koreans believe that Rhus will improve their health. However, they don't known its adverse effects, and there is no experimental report on this topic. OBJECTIVE: The principle objective of this study was to gather evidence on the toxicity and negative effects of Japanese lacquer through an experimental study with rats. METHODS: A total of 60 Sprague-Dawley rats were included in the study; 30 rats (experimental group) were given 0.1 ml of Japanese lacquer sap extract, water and food, and 30 rats (control group) were given only food and water. The WBC, the Hgb, ALT, AST, BUN and creatinine levels, the presence of pruritis and skin eruption, the skin biopsy findings and the liver biopsy findings were all checked at 5, 10, and 15 days. RESULTS: The rats that were given Japanese lacquer sap showed a higher WBC and higher ALT and AST levels and only these rats demonstrated erythroderma (5th day: 30%, 10th day: 40%, 15th day: 70%). In addition, they exhibited frequently scratching behavior. On histological examination of the skin of the rats given Rhus, dermal edema and mild inflammatory cell infiltration were found. On histological examination of the liver of the rats given Rhus, there was no significant change as compared with the rats that weren't given lacquer. CONCLUSION: In this experimental study, it was proven that Rhus could induce pruritis, erythroderma, skin inflammation and hepatotoxicity.
Animals ; Asian Continental Ancestry Group ; Biopsy ; Chickens ; Creatinine ; Dermatitis, Contact ; Dermatitis, Exfoliative ; Eating ; Edema ; Exanthema ; Hepatitis ; Humans ; Inflammation ; Lacquer ; Liver ; Pruritus ; Rats ; Rats, Sprague-Dawley ; Rhus ; Skin ; Water

A 32-year-old woman had a brownish hard plaque with localized hypertrichosis. She had fallen down some stairs 1 year previously, and the injury had left a scar on her right shin. Even though pain and the scar had gone, the brown patch had remained. During that time hair had been grown on the site. Histologic examination showed lobular panniculitis, fat necrosis, fibrotic change and lipomembranous change. We diagnosed the patient as a traumatic panniculitis with localized hypertrichosis, based on the trauma history, skin lesion and histologic findings. We report a case of traumatic panniculitis with localized hypertrichosis.
Adult ; Cicatrix ; Fat Necrosis ; Female ; Hair ; Humans ; Hypertrichosis ; Panniculitis ; Skin

BACKGROUND: Several different kinds of drugs have been used to treat chronic oral lichen planus (OLP). During the last decade, there have been several reports demonstrating success with levamisole and low dose prednisolone therapy for treating OLP. However, some OLP patients who have underlying diseases such as diabetes, hypertension and malignancy are unable to take steroids. OBJECTIVE: The aim of this study was to evaluate levamisole monotherapy for treating OLP. METHODS: Eleven patients who had OLP were treated with levamisole between 2005 and 2007. The levamisole was administered at a dose 50 mg thrice daily for three consecutive days, but then it was not administered on the following four days. RESULTS: After 2 weeks of treatment, 8 patients reported a partial response, 3 patients reported no response and no patients reported clearance of lesion. After 4 weeks of treatment, 6 patients reported a partial response, 3 patients reported no response and 2 patients reported clearance of lesion. Furthermore, after 3 months of treatment, 3 patients reported a partial response, 3 patients reported no response and 5 patients reported complete clearance of lesion. Clinical improvement was shown in 2 weeks, whilst the mean duration to achieve clearance of lesion was 6.2 weeks. Although 1 patient had mild itching, there were no significant adverse effects. CONCLUSION: Levamisole monotherapy could be a successful and safe treatment option for patients with chronic OLP and who cannot take steroids.
Humans ; Hypertension ; Levamisole ; Lichen Planus, Oral ; Prednisolone ; Pruritus ; Steroids

Mucoepidermoid carcinoma is a rare disease that usually occurs in the salivary gland. In extremely rare cases, it can originate from the skin. These cases are more aggressive than the usual mucoepidermoid carcinoma, so they have high mortality and high recurrence rates. Herein we report a case of primary cutaneous mucoepidermoid carcinoma on the cheek. A 34-year-old man had a 1.0x1.0 cm sized cystic lesion. This lesion displayed central erosion and oily discharge. He had been operated on 4 times at a private hospital, but the lesion had recurred. Histopathologically, there were many tumor islands that consisted of mucoid cells, epidermoid cells, intermediate cells and clear cells. The tumor islets showed mild positivity for Alcian blue (pH 2.5), PAS, and d-PAS stain. There was no parotid tumor on the neck-CT scan, nor on positron emission tomography. We therefore diagnosed this cheek lesion as primary cutaneous mucoepidermoid carcinoma, and it was removed completely.
Adult ; Alcian Blue ; Carcinoma, Mucoepidermoid ; Cheek ; Hospitals, Private ; Humans ; Islands ; Positron-Emission Tomography ; Rare Diseases ; Recurrence ; Salivary Glands ; Skin

Chrome may have a corrosive, necrotizing effect on tissue, so repeated skin contact to chromate might induce perforations or ulcers on the skin. This condition is called a "chrome ulcer" or "chrome hole". It appears as a small, punched-out mark with a rolled-out border. It is rarely associated with pain, but a severe itching sensation is common. We report a case of a 33-year-old man who presented with a 3 month history of several ulcers on his abdomen. He was employed at a chromate alloying process. He had several hole-like atrophic scars and unbearable pruritus. After changing his occupation, plus treatment with oral corticosteroids and antihistamines, the lesions were healed and the itching sensation faded away. However, atrophic scarring remained.
Abdomen ; Adrenal Cortex Hormones ; Adult ; Cicatrix ; Histamine Antagonists ; Humans ; Occupations ; Pruritus ; Sensation ; Skin ; Ulcer*

Chronic exogenous steroid therapy can result in hypothalamic-pituitary-adrenal axis dysfunction; this usually results from systemic steroid therapy, and is rarely associated with topical steroid therapy. We report a case of suspected iatrogenic Cushing syndrome that developed after the treatment of annular pustular psoriasis. The patient applied clobetasol-17-propionate cream (Betabate(R)) 15~30 g daily, for 4 months, without a dermatologist's prescription. After 4 months of application, the following symptoms developed; a moon face; facial flushing; hypertrichosis; central obesity; purple striae; and multiple erythematous scaly annular patches. The development of Cushing syndrome from exogenous corticosteroid treatment was supported by the typical clinical manifestations and the low basal state of the blood cortisol level, which was perhaps due to secondary suppression of the hypothalamic pituitary-adrenal axis. Steroid-containing drugs, especially during childhood, should be used with caution and monitored carefully.
Axis, Cervical Vertebra ; Cushing Syndrome* ; Flushing ; Humans ; Hydrocortisone ; Hypertrichosis ; Obesity, Abdominal ; Prescriptions ; Psoriasis

Vitiligo and morphea are two distinct entities of unknown etiology, although their existence implies that the immune system and/or the central or peripheral nervous system has been incriminated. Only a few reports of their simultaneous occurrence are on record. Here we report a case of a 39 year old woman affected with both vitiligo of the face, chest and hand and morphea of shoulder and trunk. Since the two diseases appeared after bone marrow transplantation, if could be deduced that there is a possible association between them. Two diseases observed simultaneously in this patient may be derived from the common pathomechanism. An autoimmune etiology is thought to play a part in both of these diseases.
Adult ; Bone Marrow Transplantation* ; Bone Marrow* ; Female ; Hand ; Humans ; Immune System ; Peripheral Nervous System ; Scleroderma, Localized ; Shoulder ; Thorax ; Vitiligo

Lymphangioma is an uncommon, benign hyperproliferation of the lymphatic vessels, and this consists of dilated lymphatic channels lined by a normal, single layer of endothelial cells. Various methods have been tried for the treatment of lymphangioma, including cryotherapy, electrocauterization, surgical excision, laser therapy, radiotherapy and itralesional injection of sclerosing agents. Yet these methods have shown low success rates and various side effects. It has recently been reported that intralesional injection of OK-432 is a safe and effective alternative therapy for lymphangioma, and especially for macrocystic lesion. In this report, we describe a patient with lymphangioma on the upper labial mucosa and this patient was successfully treated with intralesional injection of OK-432 without any serious complications. To the best of our knowledge, this is the first such case reported on in the Korean dermatological literature.
Cryotherapy ; Endothelial Cells ; Humans ; Injections, Intralesional ; Laser Therapy ; Lymphangioma ; Lymphatic Vessels ; Mucous Membrane ; Picibanil ; Sclerosing Solutions ; Sclerotherapy

Hyperpigmentation caused by medication or toxic agents accounts for 10~20% of all the cases of acquired hyperpigmentations. Nonsteroidal anti-inflammatory drugs, antimalarials, amiodarone, cytotoxic drugs, tetracyclines, heavy metals and psychotropic drugs are most commonly responsible for hyperpigmentation. A 74-year-old man who had taken antituberculosis drugs (rifampin and isoniazid) for 4 months developed generalized hyperpigmentation. The histopathologic finding revealed an increased number of dermal melanophages with pigment incontinence. Eight months after termination of the antituberculosis medication, his skin lesion improved without any treatment. To the best of our knowledge, this is the first reported case of generalized hyperpigmentation due to rifampin and isoniazid in a patient without adrenal insufficiency in the dermatological literature.
Adrenal Insufficiency ; Aged ; Amiodarone ; Antimalarials ; Humans ; Hyperpigmentation ; Isoniazid ; Metals, Heavy ; Psychotropic Drugs ; Rifampin ; Skin ; Tetracyclines