Intravenous immunoglobulin-induced, non-eczematous, vesiculobullous eruptions in Stevens-Johnson syndrome.

Intravenous immunoglobulin (IVIG) has emerged as a promising treatment that interrupts the progression of Stevens-Johnson syndrome (SJS). Our patient experienced an uncommon adverse effect, non-eczematous, vesiculobullous eruptions, after treatment with IVIG. These new lesions developed rapidly on the palms while most previous SJS bullous lesions subsided. A skin biopsy of these new lesions showed an intracorneal vesicle, without epidermal necrosis, with inflammatory cell infiltration. IVIG-induced, vesiculobullous eruptions are discussed, along with their possible pathogenesis. With the increasing use of IVIG for treatment of bullous dermatoses, recognition of this rare adverse effect is important for prompt differential diagnosis.

Cutaneous normolipemic plane xanthoma with supraglottic involvement in a patient with hand-Schüller-Christian Disease: a case report.

Normolipemic plane xanthoma (NPX) is a histiocytic disorder characterized by yellow-orange plaques in the periorbital areas, neck, upper trunk, and flexural folds. Association with systemic disease or paraproteinemia has been reported previously, but rarely with Langerhans cell histiocytosis (LCH). We report a case of Hand-Schüller-Christian disease (a type of LCH) in a patient who developed NPX with supraglottic involvement. NPX developed after several courses of chemotherapy and the supraglottic xanthoma occurred about 2 years later. The coexistence of LCH and non-LCH histiocytic lesions in this patient could be a result of chemotherapy-induced changes or may be just coincidental.

Bullous pemphigoid in a renal transplant recipient: a case report and review of the literature.

Bullous pemphigoid (BP) is an autoimmune disease with chronic, recurrent bullous eruptions. BP has been reported to be associated with drugs, physical stimuli, malignancies, and immune abnormalities. Its association with renal transplant is rare and only four cases have been reported. We present a case of BP in a 52-year-old man with chronic hepatitis B and C infection who underwent a cadaveric renal transplant 13 years earlier. His graft was still functioning well when BP appeared. The occurrence of BP in our patient might be a result of drugs (furosemide or tacrolimus), viruses, or renal allograft. As the patient was receiving regular T-cell immunosuppressant therapy, his BP lesions were recalcitrant to corticosteroid treatment. We discuss the pathogenesis and treatment of such patients.

Bilateral sporotrichoid lymphocutaneous dermatosis in a drug abuser: case report and review of the literature.

Bilateral sporotrichoid mycobacterial cutaneous infection is unusual and has been reported in only three cases involving different species and with an unknown route of infection in the literature.We report a case of bilateral sporotrichoid dermatosis in an immunocompetent patient with a history of intravenous heroin injection before development of the skin lesions. Both special stain and culture of biopsy specimen were negative. Finally, Mycobacterium fortuitum was identified by a polymerase chain reaction-based method. The patient responded well to clarithromycin and ciprofloxacin therapy. This case represents an unusual primary cutaneous M. fortuitum infection manifested as bilateral sporotrichoid lesions of the limbs. Review of previous reported cases of bilateral sporotrichoid mycobacterial infection shows different isolated organisms and routes of infection to that found in our case.

Clinical manifestations and characteristics of patients with acute generalized exanthematous pustulosis in Asia.

Acute generalized exanthematous pustulosis is a rare, sterile inflammation of the skin (and mucous membranes) characterized by acute onset of generalized pustule formation, fever and leukocytosis, and a rapid resolution. A computer-based search of the literature revealed only a limited series of reports, none of them based on Asian patients. We identified and retrospectively analysed 16 biopsy-proven and criteria-matched patients over a 15-year period in a single medical centre. The study showed a female predominance (11 of 16 patients), a relatively low association with systemic drugs (62.5% vs. 87% in a previous report) and normal renal function (in contrast to a previous report where 32% of patients had renal failure). The study also revealed high levels of C-reactive protein (76.0 mg/l) and normal absolute eosinophil count (176.2/microl) in most patients. Furthermore, there was no difference between different treatment regimens regard-ing the course and duration of the disease or the length of fever (p>0.05). In addition, if the patients were subdivided by aetiology into those strongly associated with or not strongly associated with systemic drugs, a significant difference was found in age of onset between the 2 groups (p<0.01).

Metastasis of mediastinal epithelioid angiosarcoma to the finger.

Epithelioid angiosarcoma (EA) is a rare malignant, vascular tumor that is usually observed in middle-aged and elderly males. Cutaneous metastasis of EA is extremely rare. We report the case of a 41-year-old woman presenting with a painful bluish, bulla-like lesion on the distal extent of the left third finger. The patient had recently been diagnosed with mediastinal EA with disseminated metastases. The skin biopsy specimen revealed metastatic EA. This is thought to be the first reported metastasis of EA to the finger. Whenever a patient has metastatic disease, acrometastases should be considered in the differential diagnosis of inflammatory lesions of the digits and a skin biopsy should be performed.

Indigenous leishmaniasis in Taiwan: report of a case.

Taiwan is not considered an endemic area of leishmaniasis. Imported cases are encountered infrequently, and only two cases of indigenous cutaneous leishmaniasis have been reported.(1) We found one new case in the past 20 years. The patient presented with erythematous plaques on the nasal bridge and right thumb. Skin biopsy specimens from both sites revealed numerous Leishman-Donovan bodies in macrophages. There was no history of travel outside the country, and the diagnosis of indigenous cutaneous leishmaniasis was made. Polymerase chain reactions (PCR) identified the species as Leishmania tropica. The route of infection in this patient is unclear. Because pentavalent antimony, the drug of choice for leishmaniasis, is not available in Taiwan, the patient was treated with levamisole and potassium iodide, with an excellent response.

A birthmark survey in 500 newborns: clinical observation in two northern Taiwan medical center nurseries.

BACKGROUND: The prevalence of birthmarks in Taiwanese newborns has not been well studied. How gender and maturity of newborns affect presentation and prevalence of vascular birthmarks, in particular, has not been well documented. The aim of this survey was to catalog the above-mentioned problems. METHODS: From May 9 through July 14, 2000, 500 newborns in Linkou and Taipei Chang Gung Memorial Hospital nurseries were examined for the presence of birthmarks. The data were collected and stored in Excel software and analyzed using chi2-test and Fisher's exact test for statistical association. RESULTS: The most common pigmentary birthmark was the Mongolian spot (61.6%), followed by congenital melanocytic nevi (0.6%), café-au-lait spots (0.4%), and nevi depigmentosus (0.4%). Vascular birthmark frequency was: salmon patch (27.8%), Port-Wine stain (0.6%), and hemangioma (0.2%). Other miscellaneous changes included preauricular sinus (0.6%), skin tag (0.6%), and accessory auricle (0.2%). Prevalence of the vascular salmon patch birthmarks in female infants and male infants was 31.0% and 26.6%, respectively, but the difference was not statistically significant (p = 0.51). The prevalence of vascular salmon patch birthmarks in full-term infants was 28.0%, and 25.8% in preterm infants. However, there was also no statistical difference (p = 0.96) between the two. The two nevus depigmentosus cases were isolated types and neither of these were distributed along the Blaschko lines. CONCLUSIONS: The Mongolian spot was the most common birthmark in Taiwanese newborns, followed by the salmon patch. All of the other birthmarks had prevalence of less than 1%. The prevalence of vascular salmon patch birthmarks was noted in 31.0% of female infants and in 26.6% of male infants. The prevalence of vascular salmon patch birthmarks in full-term infants was 28.0% versus 25.8% in preterm infants. However, the findings regarding vascular birthmarks were not statistically significant between the differing gender and maturity groups.

Bullous amyloidosis in a hemodialysis patient is myeloma-associated rather than hemodialysis-associated amyloidosis.

We report a 78-year-old woman on hemodialysis who presented with refractory multiple pruritic vesicles and bullae on her trunk and extremities for 2 months. Histopathologic examination of skin biopsy specimen showed subepidermal bullae with many amyloid deposits in the papillary dermis. No evidence of systemic amyloidosis could be found on physical examination. While the initial clinical diagnosis was bullous pemphigoid, the histopathology and direct immunofluorescence result favored hemodialysis-associated amyloidosis. However, immunochemical study for beta(2)-microglobulin was negative. Further hematologic and immunologic work-up revealed the presence of multiple myeloma and that the deposit was AL amyloid. This is the first case of bullous amyloidosis in a hemodialysis patient and should remind dermatologists that bullous amyloidosis should be considered in addition to the usual presentation of porphyria cutanea tarda and pseudoporphyria for bullous dermatosis in the hemodialysis patient. We also suggest that hemodialysis-associated amyloidosis should not be taken for granted in the hemodialysis patient with cutaneous amyloidosis without systemic signs and symptoms. Further testing for other types of amyloid should be performed.

Urachal duct remnant-like umbilical clear cell acanthoma in an infant: an unusual presentation and pitfall in clinical practice.

BACKGROUND: Although an umbilical nodule is common in neonates and young infants, an umbilical nodule of poor therapeutic response will increase the likelihood of other uncommon etiology. Clear cell acanthoma (CCA) has never been described as an oozing umbilical nodule on infants. METHODS: To present a case of CCA which occurred on an 8-month-old female infant presenting with one weeping nodule on the umbilicus since early neonate. The lesion underwent skin biopsy and subsequent hematoxylin-eosin and periodic acid-Schiff staining. RESULT: Three courses of liquid nitrogen cryosurgery were performed after the diagnosis of CCA was confirmed. The lesion resolved rapidly. CONCLUSION: This report presents the first and youngest case of umbilical CCA in the English literature. This case supports the inflammatory dermatosic nature of CCA.

Treatment of a tufted angioma with intense pulsed light.

Tufted angioma is a rare cutaneous angiomatous proliferation named because of its characteristic histologic pattern of grouped dermal capillary tufts. Lesions usually begin in infancy or early childhood but rarely are congenital. Clinical manifestations are dull red coalescent papules and plaques, most commonly located on the neck, shoulders, and upper back, and can sometimes be tender. Although transformation to malignancy has not been described, tufted angiomas do not tend to regress. Effective treatments reported in the literature are scarce. We report an adult case of tufted angioma, with unusual presentation as annular plaques, which was alleviated after treatment with intense pulsed light in terms of both cosmetics and discomfort.

Malignant cutaneous tumors of the scalp: a study of demographic characteristics and histologic distributions of 398 Taiwanese patients.

BACKGROUND: The scalp is a unique anatomic region, in which pilosebaceous follicles are concentrated. OBJECTIVE: We sought to investigate demographic characteristics and histologic distributions of malignant scalp tumors. METHODS: Primary and metastatic scalp malignancies diagnosed histopathologically between 1983 and 2003 were reviewed. Age at diagnosis, sex, and histologic types were analyzed. RESULTS: A total of 398 Taiwanese patients (200 males, 198 females) were selected. Age at diagnosis ranged from 3 to 103 years. Most malignant scalp tumors (69.8%) occurred in those 50 years or older. Basal (41.2%) and squamous (16.6%) cell carcinomas were the most common histologic types. Surprisingly, metastatic tumors (12.8%) came in third, in which lung cancers were the most frequent primary tumor in both male and female patients. LIMITATIONS: In our series, the case number of metastatic scalp malignancies was underestimated because not all patients with metastatic scalp tumors received a scalp skin biopsy. CONCLUSION: Because a wide spectrum of primary and metastatic malignancies can occur on the scalp, scalp inspection should be included in general screening for either skin or internal cancers.

Cellulitis-like cutaneous metastasis of uterine cervical carcinoma.

Metastasis to the skin from cervical carcinoma is relatively uncommon. Herein we present a 41-year-old woman with a history of cervical carcinoma with severe facial erythematous swelling and telangiectasia. She was initially treated for cellulitis without improvement. A skin biopsy specimen revealed widespread intravascular tumor emboli in the dermis and subcutis, resembling the so-called inflammatory carcinoma of the breast. As this is an unusual clinical presentation for the metastasis of cervical carcinoma, this case is reported.

Warm and neutral tumescent anesthetic solutions are essential factors for a less painful injection.

BACKGROUND: Tumescent local anesthesia is widely used in dermatologic surgery. Minimizing pain associated with injections is crucial to successful surgical procedures. OBJECTIVE: This study investigates the pain associated with warm and room temperatures in neutralized or nonneutralized tumescent anesthetic solutions injection. METHODS: Thirty-six patients with axilla osmidrosis who underwent local anesthesia for surgery were randomly assigned to three groups. Group A received warm neutral (40 degrees C) and room-temperature neutral (22 degrees C) tumescent injections to each axillary region. Group B received warm neutral (pH 7.35) and warm nonneutral (pH 4.78) tumescent injections on each side of axilla. Group C received warm nonneutral and room-temperature nonneutral tumescent injections on each side of axilla. Pain associated with infiltration of anesthesia was rated on a visual analog scale (VAS). RESULTS: A statistically significant decrease (p < .001) in pain sensation was reported on the warm, neutral injection side (mean rating, 32.7 mm) compared with the room-temperature, neutral injection side (mean rating, 53.3 mm). Patient-reported pain intensity was significantly lower on the side that received warm, neutral tumescent anesthesia (mean rating, 26.8 mm) than on the side receiving warm, nonneutral tumescent anesthesia (mean rating, 44.9 mm; p < .001). The difference in VAS scores between warm neutral (mean rating, 23.9 mm) and room-temperature nonneutral (mean rating, 61.2 mm) was statistically significant (p < .001). CONCLUSION: The warm, neutral tumescent anesthetic preparation effectively suppressed patient pain during dermatologic surgical procedures.

Early congenital syphilis and erythema multiforme-like bullous targetoid lesions in a 1-day-old newborn: detection of Treponema pallidum genomic DNA from the targetoid plaque using nested polymerase chain reaction.

A 1-day-old male newborn was born with respiratory distress, low birth weight, hepatosplenomegaly, and bullous targetoid skin lesions over the face, back, buttocks, and extremities. A diagnosis of early congenital syphilis was made based on a treponemal serologic test. Pathologic examination of the skin lesion showed scattered dyskeratotic cells in the epidermis and interface dermatitis consistent with erythema multiforme. No spirochete could be found in the skin sections staining with Warthin-Starry stain. Using nested polymerase chain reaction, treponemal genomic DNA fragments encoding DNA polymerase I were detected.