Angiolymphoid Hyperplasia with Eosinophilia That Was Possibly Induced by Vaccination in a Child

Angiolymphoid hyperplasia with eosinophilia (ALHE) is a rare benign vasoproliferative disease of an unknown cause involving the skin or subcutaneous tissue of the head and neck, and particularly around the ear. It predominantly affects Caucasian adults during the third and fourth decades and it very rarely occurs in children. We experienced a case of ALHE in a 2-year-old Korean boy who had a firm, pruritic, skin-colored, subcutaneous nodule on his right upper arm. The histopathological findings were compatible with ALHE and they showed prominent vascular changes with epitheloid or histiocytoid endothelial cells surrounded by inflammatory cells, including a large proportion of eosinophils. This unusual distribution of the lesion and the young age of the patient may be associated with vaccination.

Expression of N-terminal truncated desmoglein 3 (Delta NDg3) in epidermis and its role in keratinocyte differentiation

During a search for keratinocyte differentiation-related genes, we obtained a cDNA fragment from the 5'-untranslated region of a previously identified splicing variant of desmoglein 3 (Dg3). This transcript encodes a protein of 282 amino acids, which corresponds to the N-terminal truncated intracellular domain of Dg3 (Delta NDg3). Northern blot analysis detected a 4.6-kb transcript matching the predicted size of Delta NDg3 mRNA, and Western blot analysis with an antibody raised against the Dg3 C-terminus (H-145) detected a 31-kDa protein. Increased Delta NDg3 expression was observed in differentiating keratinocytes by RT-PCR and Western blot analysis, suggesting that Delta NDg3 is indeed a differentiation-related gene product. In immunohistochemical studies of normal and pathologic tissues, H-145 antibody detected the protein in the cytoplasm of suprabasal layer cells, whereas an antibody directed against the N-terminal region of Dg3 (AF1720) reacted with a membrane protein in the basal layer. In addition, Delta NDg3 transcript and protein were upregulated in psoriatic epidermis, and protein expression appeared to increase in epidermal tumors including Bowen's disease and squamous cell carcinoma. Moreover, overexpression of Delta NDg3 led to increased migration and weakening of cell adhesion. These results suggest that Delta NDg3 have a role in keratinocyte differentiation, and that may be related with tumorigenesis of epithelial origin.

A Case of Isolated Plexiform Neurofibroma in a Patient with Myasthenia Gravis

We report a case of an isolated plexiform neurofibroma occurring in a patient with myasthenia gravis. A 48-year-old man presented with asymptomatic skin-colored nodules on the tip of his 4th finger. Microscopically, a plexiform neurofibroma was identified located in the dermis that appeared to originate from small superficial nerves. He had a 20-year history of treated myasthenia gravis; otherwise, his personal and family histories were unremarkable. Given that myasthenia gravis is a disorder of the peripheral nerves, plexiform neurofibromas could be associated with myasthenia gravis. However, the development of an isolated plexiform neurofibroma in a case of myasthenia gravis has not yet been reported. The occurrence of a neurofibromas in a patient with myasthenia gravis suggests a link in the pathogenesis of these two diseases.

A Clinical Study of 263 Patients with Childhood Vitiligo / 대한피부과학회지

BACKGROUND: Vitiligo is an acquired pigmentary disorder and half of these patients experience disease onset during childhood. The psychosocial complications of vitiligo can be substantial, and especially for children. There has been no study that has compared the clinical features according to the symmetry of the vitiligo lesions of patients with childhood vitiligo. OBJECTIVE: The purpose of this study is to evaluate the clinical aspects and distribution of the vitiligo lesions according to the symmetry in patients with childhood vitiligo. METHODS: The clinical characteristics and distribution of the vitiligo lesions according to the symmetry were investigated in the 263 childhood patients who were diagnosed with vitiligo at the Dermatology Department of our University Hospital from January 1991 to August 2008. RESULTS: Of the 263 patients, 133 (50.6%) had the asymmetric vitiligo lesions, 76 (28.9%) had the symmetric lesions and 54 (20.5%) had the multiple lesions. Of the patients with asymmetric vitiligo lesions, 91 (34.6%) had the localized lesions and 42 (16.0%) had the segmental lesions. The most common site of the vitiligo lesions was the face (54.8%). In case of the vitiligo lesions on the scalp, face and neck, the asymmetric type was predominant. The lesions on the scalp and the face showed a lesser tendency to spread to other sites. CONCLUSION: Childhood vitiligo showed the higher prevalence of asymmetric vitiligo lesions. It is expected that the asymmetric type of childhood vitiligo, and especially that on the scalp and the face, may have a favorable clinical course.

A Case of Bullous Lichen Sclerosus et Atrophicus / 대한피부과학회지

Lichen sclerosus et atrophicus (LSA) is a benign, chronic inflammatory dermatosis that usually affects the female genitalia. It may also involve any other areas, and it is rarely seen on the face, oral mucosa and palm and sole. The lesion of LSA primarily occurs as a whitish or yellowish papule, and later it becomes a well-defined plaque. However, bullous LSA, which has a localized or generalized form, occurs as a rare variant of LSA. The exact prevalence of bullous LSA is uncertain. It generally involves not only the genitalia, which is vulnerable to trauma, but also extragenital sites. We present here a case of extragenital LSA with bullae on the upper back of a 33-year-old female. Histologic examination revealed hyperkeratosis, atrophy, vacuolar alteration of the basal layer and homogenization of the collagen in the upper dermis. In addition, increased collagen bundles and lymphocytic infiltration around the vessels in the lower dermis were also noted.

Localized Scleroderma-like Reaction Induced by Doxifluridine / 대한피부과학회지

Exogenous factors, including environmental substances and drugs, are known to induce scleroderma-like reactions. Various scleroderma-like reactions induced by anti-cancer drugs have recently been reported. This is the first report that doxifluridine (Didox), an oral prodrug of the antineoplastic agent 5-fluorouracil (5-FU), induced a localized sclerderma-like reaction. A 51-year-old woman was referred to our clinic with multiple pearly, shiny patches on both her breasts, her pelvis and her back. After surgical excision and radiation therapy due to her right breast cancer, she took Didox for 7 months. A skin biopsy specimen revealed that the dermal collagen thickening extended even to the subcutaneous tissue. The routine laboratory tests were within the normal ranges and the tests for antinuclear antibody (ANA), anti SS-A antibody, anti SS-B antibody and anti U1-RNP antibody were all negative. After discontinuation of Didox, the lesions gradually improved. Based on these finding, we diagnosed this case as a localized scleroderma-like reaction induced by doxifluridine and we should pay attention to detect this adverse effect of the long term use of doxifluridine.

Clinical Trial to Evaluate the Safety and Efficacy of Scalp Med(R) in Treatment of Patients with Androgenetic Alopecia / 대한피부과학회지

BACKGROUND: Androgenetic alopecia (AGA) is characterized by the structural miniaturization of androgen-sensitive hair follicles in susceptible individuals and is anatomically defined within a given pattern of the scalp. Oral finasteride and topical minoxidil are the only drugs whose effect and safety were demonstrated and approved by the FDA. However, the treatment result of these medications are not always satisfactory. Here, we report the result of a clinical trial of the new topical agent, Scalp med(R) to patients with AGA. OBJECTIVE: The goal of this study was to test Scalp med(R), the solution including minoxidil, retinol, polysorbate 80 and phytosterol in the treatment of AGA. METHODS: Included in this study were males between the ages of 20 and 60 years, in good health, with mild to moderate AGA. Efficacy was evaluated by terminal hair count per 1 cm2 and hair thickness. RESULTS: The results of this clinical trial showed a highly positive response to treatment. After 24 weeks of treatment, a significant improvement in hair count and hair thickness was demonstrated in all the patients. CONCLUSION: This clinical study establishes the effectiveness and safety of Scalp med(R) in the treatment of AGA.

A Case of Amniotic Band Syndrome / 대한피부과학회지

The amniotic band syndrome is a collection of fetal malformations associated with fibrous band that appears to entangle or entrap various fetal part, in utero, leading to deformation, malformation or disruption. Its pathogenesis has been debated in the literature for many years. The associated anomalies vary from minor limb anomalies to major craniofacial defects and visceral defects. We experienced a case of limb anomalies due to amniotic band and present the findings with a brief review of literature.

Expression of Neutrophil Gelatinase-Associated Lipocalin in Calcium-Induced Keratinocyte Differentiation

In a previous search for the differentially expressed genes in keratinocyte differentiation, we identified neutrophil gelatinase-associated lipocalin (NGAL) as a calcium- induced gene. In this study, we further verified the expression of NGAL in cultured keratinocytes as well as in several skin diseases. Reverse transcription-polymerase chain reaction (RT-PCR), Western blot, and ELISA clearly showed that NGAL expression was markedly increased in calcium-induced keratinocyte differentiation in vitro. However, in our previous report, NGAL expression was not detected in normal skin tissue except for hair follicle by in situ hybridization and immunohistochemistry, indicating the difference of cell status between in vitro and in vitro conditions. Interestingly, NGAL expression was highly increased in psoriasis-like inflammatory disorders (lichen planus and pityriasis rubura pilaris) and skin cancers (keratoacanthoma and squamous cell carcinoma), implying that NGAL may be related with the epidermal hyperplasia. Collectively, these results reveal the potential importance of NGAL in the maintenance of skin homeostasis.

Quality of Life of Alopecia Areata Patients / 대한피부과학회지

BACKGROUND: Hair constitutes an integral part of our self and our self-identity, which is why hair loss may cause a broad range of psychological problems related to our identity. However, the association between quality of life (QOL) in alopecia areata (AA) patients and their clinical characteristics has not yet been investigated in Korea. OBJECTIVE: The purpose of this study is to compare the QOL in patients with AA and healthy controls by using Hairdex scale. The relationship between their QOL and their clinical characteristics will be investigated. METHODS: 102 AA patients and 82 healthy controls were recruited in this study and compared by use of Hairdex. Correlation analysis was performed to examine the relationship between the QOL and the clinical characteristics of patients with AA. RESULTS: The total Hairdex scores and all domain scores, except those of the symptomatic domain, of the Hairdex scales in AA were higher than those of the healthy controls. Clinical characteristics, including the age, disease duration, AA type, AA area, treatment satisfaction, treatment history, anxiety and depression, showed significant correlation with QOL. Of these characteristics, the depression and treatment satisfaction showed strong association with QOL. CONCLUSION: AA can a impair patient's quality of life and has a significant psychological impact.

Assessment of the Quality of Life in Vitiligo Patients / 대한피부과학회지

BACKGROUND: Although vitiligo does not cause direct physical impairment or life-threatening conditions, it can produce significant psychosocial problems. Nevertheless, its effect on the psychosocial well-being of patients has been neglected. OBJECTIVE: This study compared quality of life (QOL) in patients with vitiligo and in healthy controls, and assessed the impact of vitiligo on the patients' QOL using Skindex-29. METHODS: This study recruited 105 vitiligo patients and 105 healthy controls, and the QOL scores of the groups were compared using Skindex-29. Statistical analyses were performed to examine the relationship between QOL and clinical characteristics. RESULTS: The total Skindex-29, emotion, and function scores, but not the symptom scores, were significantly higher in the vitiligo group than in the healthy controls. QOL, including symptom, function, and emotion scores, was significantly impaired in women, the elderly, married patients, and patients with a longer duration of disease, greater area of vitiligo, and lesions appearing on the exposed sites. But, symptom scores generally did not differ significantly. All aspects of QOL were impaired in patients with a previous treatment history, longer duration of treatment, and relatively low levels of satisfaction with treatment. CONCLUSION: Vitiligo can impair a patient's QOL. Therefore, it is important for the dermatologist to recognize and manage psychological problems to improve the QOL of vitiligo patients.

A Case of the Generalized Type of Acquired Dermal Melanocytosis with ABNOM and Acquired Bilateral Nevus of Ito-like Macules / 대한피부과학회지

Dermal melanocytosis is histologically characterized by the presence of ectopic melanocytes in the dermis. Mongolian spots, nevus of Ota, nevus of Ito and blue nevus are the most common types and these are usually present at birth or in early childhood. However, it has been reported that several types of dermal melanocytosis could appear in adult life. We report here on an unusual case of acquired dermal melanocytosis occurring in a 51 year-old female. She had bilaterall brownish or slate-bluish pigmented patches on the face and the posterior auricular, supraclavicular, scapular and back areas. The skin biopsy specimen taken from the upper back revealed scattered, darkly pigmented, spindle-shaped cells and dendritic cells containing abundant golden brown pigment in the dermis.

The Clinical Significance of Symmetry in the Distribution of Vitiligo Lesions / 대한피부과학회지

BACKGROUND: Vitiligo is an acquired disease that causes depigmented patches of various sizes. The most widely used method of classification is by the distribution, which is categorized into four types: localized, segmental, universal, and generalized vitiligo. In addition, vitiligo can be divided into segmental and non-segmental, according to its relation to dermatomes. However, few studies have compared the clinical features and prognosis with the symmetry of the vitiligo lesions. OBJECTIVE: We examined 690 vitiligo patients to investigate the clinical significance of symmetry in the distribution of vitiligo lesions. METHODS: The clinical characteristics, distribution, and lesion sizes were investigated in 690 patients who were diagnosed with vitilgo at the Dermatology Department of Chungnam National University Hospital from January 1991 to August 2006. RESULTS: Of the 690 vitiligo patients, 369 (53.3%) had symmetric vitiligo lesions and 321 (46.5%) had asymmetric lesions. Patients with symmetric lesions had an older age of onset, longer duration of disease, wider distribution area, and greater tendency of the lesions to enlarge with time. CONCLUSION: Our clinical classification method, based on the the symmetry of the vitiligo lesions, may help physicians to predict the course and prognosis of the disease. Therefore, the dissemination of new vitiligo lesions in symmetric vitiligo patients should be observed closely.

A Case of Trichobalstic Carcinoma / 대한피부과학회지

Trichoblastoma is a benign cutaneous neoplasm that consists of follicular germinative cells. There have only been a few case reports describing the malignant counterpart of trichoblastoma. We report trichoblastic neoplasms that show histological features of malignancy. The histological sections revealed locally aggressive neoplasms showing prominent differentiation towards hair germ and follicular sheath with formation of papillary mesenchymal bodies and follicular bulb-like structures. The tumor was widely excised with a clear margin. No sign of recurrence has been noted 1 year later.

The Efficacy of Autologous Epidermal Grafting Based on Clinical Types for the Treatment of 320 Stable Vitiligo Lesions / 대한피부과학회지

BACKGROUND: Autologous epidermal grafting was reported by several authors to be successfully used for the treatment of stable vitiligo lesions. However, there have been only a few studies with a sufficient number of cases and long-term follow up. OBJECTIVE: The purpose of this study was to evaluate the long-term efficacy of autologous epidermal grafting for the treatment of stable vitiligo lesions. METHODS: We evaluated 187 patients (320 lesions) after autologous epidermal graftings using suction blistered epidermis. The effects were analyzed based on age, sex, clinical types, recipient sites, the ablative methods for recipient sites and the time after the operation. RESULTS: Autologous epidermal grafting was more effective for younger patients, for asymmetrical types and when the recipient sites were the head and neck. Pigment production increase until about 3 years after the operation was also found for patients who were evaluated regularly for more than 3 years. CONCLUSION: Autologous epidermal grafting is a safe, effective treatment modality for stable vitiligo. In order to determine the long-term efficacy of epidermal grafting, doctors should closely observe for at least 3 years after the grafting.

A Case of Erythema Nodosum-like Eruptions as a Manifestationof Azathioprine Hypersensitivity / 대한피부과학회지

Azathioprine is an immunosuppressant that has been widely used in the prevention of organ post-transplantation rejection, rheumatoid arthritis, inflammatory bowel disease and myasthenia gravis. The adverse effects reported with this drug have been classified as early events that include fever, hypotension, vasculitis and erythema nodosum-like eruptions or as late events that include bone marrow depression, nausea and vomiting. Although erythema nodosum-like eruptions could be useful for recognition of azathioprine hypersensitivity, this eruption has not yet been reported in Korean dermatological literature. We report, herein, a 68-year-old female with myasthenia gravis, who presented with tender, erythema nodosum-like eruptions on both upper and lower extremities 14 days after the first administration of azathioprine. Her symptoms improved 2 days after discontinuation of azathioprine and healed with no scarring change.

A Case of Acral Erythema Induced by Etoposide / 대한피부과학회지

Etoposide is a semi-synthetic podophyllotoxin that binds to microtubular proteins to inhibit cell division. It has been used extensively in the treatment of both solid and hematologic malignancies. Chemotherapy-induced acral erythema is a distinctive syndrome of painful, symmetric, well-defined swelling and erythema of the palms and soles seen in patients receiving high-dose chemotherapy. It occurs most commonly with fluorouracil, doxorubicin, and especially cytosine arabinoside. Although etoposide, mercaptopurine and methotrexate have also been implicated. Here we report a case of acral erythema induced by etoposide in a 15-year-old child with neuroblastoma.

Three Cases of Allopurinol-induced DRESS Syndrome / 대한피부과학회지

Allopurinol (4-hydroxypyrazolo-[3,4-d]pyrimidine) is an effective and widely used xanthine oxidase inhibitor administered in the treatment of hyperuricemic states such as gout. Allopurinol-induced DRESS (Drug rash with eosinophilia and systemic symptoms) syndrome is characterized by hematologic abnormalities, especially eosinophilia and mononucleosis-like atypical lymphocytosis, skin rash, fever, lymph node enlargement and single or multiple organ involvement, which starts within 8 weeks after the initiation of therapy. We report three cases of allopurinol-induced DRESS syndrome who developed erythematous skin eruption six weeks, nine weeks and seven weeks, respectively, after allopurinol therapy. The clinical, laboratory and histologic findings of these patients were compatible with allopurinol-induced DRESS syndrome.

Cutaneous Plasmacytosis Improved by PUVA Therapy / 대한피부과학회지

Cutaneous plasmacytosis is a rare disorder characterized by multiple red-brown plaques, mainly on the trunk, which histologically show a marked hyperplasia of mature plasma cells with no mitotic figures or cellular atypia. It is sometimes accompanied by extracutaneous symptoms like lymphadenopathy or interstitial pneumonia, but without plasma cell infiltration. Although many treatments such as corticosteroid lesional injection, cyclophosphamide, melphalan, systemic PUVA, and local tacrolimus application have been introduced for cutaneous plasmacytosis, there is no reported post-treatment data on cutaneous plasmacytosis in Korea. We report a case of a Korean male patient who was diagnosed with cutaneous plasmacytosis which was improved by PUVA therapy.

Neutrophilic Eccrine Hidradenitis Probably Induced by Intravenous Administration of Placental Extracts / 대한피부과학회지

Neutrophilic eccrine hidradenitis (NEH) is a rare, self-limited, neutrophilic dermatosis characterized histologically by a neutrophilic infiltration around the eccrine coils and necrosis of the eccrine glands. It occurs most commonly in patients undergoing chemotherapy for acute myelogenous leukemia and other malignant disease, but, other associations, such as infections, drugs or even in generally healthy individuals. We report a case of 45-year-old Korean woman with numerous erythematous papules on her trunk after treatment with intravenous administration of placental extracts. The hisopathological findings were compatible with neutrophilic eccrine hidradenitis.