Shigeto Yanagihara

Linear immunoglobulin A/immunoglobulin G bullous dermatosis associated with Vogt-Koyanagi-Harada disease.

Vogt–Koyanagi–Harada disease is characterized by marked bilateral uveitis associated with symmetric vitiligo, alopecia, poliosis and dysacousia. Linear immunoglobulin (Ig)A bullous dermatosis (LABD) is characterized by small, tense, subepidermal bullae caused by IgA type autoantibody targeting the basal lamina. LABD patients sometimes show coexistence of IgG type autoantibody, termed linear IgA/IgG bullous dermatosis (LAGBD). We reported a 35‐year‐old Japanese male case of combined LAGBD and Vogt–Koyanagi–Harada disease. His human leukocyte antigen typing was −A24, B52, C*1202, DR*1502, DQ*0601. Immunoblot revealed that patient sera reacted to both 180‐ and 230‐kDa proteins at the IgA and IgG level. Because Vogt–Koyanagi–Harada disease and LABD are reported to be associated with other autoimmune diseases, it is probable that Vogt–Koyanagi–Harada disease and LAGBD in our case may be associated with each other in the pathomechanism. However, we cannot exclude the possibility of this being mere coincidence.

Protective effect of hochuekkito, a Kampo prescription, against ultraviolet B irradiation‐induced skin damage in hairless mice

A Kampo prescriptions, hochuekkito (HET) has been utilized for treating functional conditions such as general fatigue, compromised state and gastrointestinal motility disorder. Recently, HET has attracted the attention of dermatologists because of its clinically positive effects in atopic dermatitis (AD) treatment. To explain this positive effect of HET, we examined its protective ability against oxidative skin stress using a murine model. The dorsal region of 8‐week‐old male HR‐1 hairless mice, which were raised on a HET (0%, 2% and 10%) mixed diet, was irradiated once with 70 mJ/cm2 of ultraviolet (UV)‐B light. After 4 days, transepidermal water loss (TEWL) and stratum corneum water content (SCWC), were determined as a measure of degree of skin dysfunction. To estimate the amount of active oxygen generated, the stratum corneum catalase activity (SCCA) and stratum corneum carbonylated protein (SCCP) content in the tape‐stripped stratum corneum samples were measured. We also measured the H2O2 scavenging ability of HET, and analyzed the changes in the expression levels of several inflammation and oxidative stress‐related genes in the skin of HET‐fed mice. In control mice, exposure to UV‐B led to significant increases in TEWL and SCCP and significant decreases in SCWC and SCCA. These UV‐B‐induced changes were reduced in mice administrated HET, and the reduction was HET dose‐dependent. Our results suggested that HET offered a protective effect against UV‐B‐induced skin damage. We also found that HET had relatively low ability to scavenge H2O2, and expression level of cyclooxygenase‐2 mRNA decreased in HET‐fed mouse.

Localization of cellular neurothekeoma with magnetic resonance microscopy Imaging

change for 7 months. Pigmented epithelioid melanocytoma usually presents as a solitary, pigmented, dome-shaped nodule and has a predilection for the extremities and trunk. However, it rarely develops on oral and genital mucosae. It is characterized by medium to large heavily pigmented epithelioid melanocytes arranged as solitary units between the collagen bundles of the dermis. It requires differentiation from tumors like Spitz nevus, blue nevus, malignant blue nevus and malignant melanoma. In contrast to PEM, Spitz nevus is sparsely pigmented, and shows nested features in the reticular dermis. Moreover, Kamino bodies are occasionally found, and immunoreactivity for HMB-45 is negative in Spitz nevus. The presence of epithelioid cells is the most characteristic feature of PEM differentiating from blue nevus, which is mostly composed of spindled or dendritic cells. Malignant blue nevus can be differentiated by its high-grade cellular atypia, brisk mitotic activity, necrosis and increased Ki-67 immunoreactivity. Malignant melanoma can be easily differentiated by its typical pagetoid activity along the dermoepidermal junction, cellular pleomorphism, significant mitotic activity, infiltrating margins and peripheral inflammation. To our knowledge, PEM occurring on the genital mucosa has been rarely reported in the published work. Herein, we report a sporadic case of PEM found on the glans penis, a rare location, of a teenage boy without clinical evidence of the Carney complex. CONFLICT OF INTEREST: None.

A Case of Anaphylaxis caused by Lemon Sorbet

A 17-year-old Japanese woman, without nasal pollen allergy, had been suffering from cashew allergy for 6 years, and lip swelling and oral irritation after eating citrus fruits for 10 years. Moreover, she experienced anaphylaxis at the age of 16; when she had eaten a lemon sorbet, played a sousaphone for 45 minutes and walked for 5 minutes, she had noticed swelling of her hands and dyspnea. She had been transferred to the hospital and taken a drip infusion of hydrocortisone, and her systemic symptoms had improved. She came to our hospital to examine whether she had risk of lemon allergy or not because she wanted to participate in the “lemon festival” held in Menton, France.

Severe dermatitis mimicking deep vein thrombosis caused by hexyldecanol

A 44-year-old man was referred to our outpatient clinic because of severe swelling of the right leg. The patient had applied an over-thecounter (OTC) cream, Berybear cream (Daiichi Sankyo Co Ltd., Tokyo, Japan), for a pruritic eruption on the neck. As the cream had no effect, he consulted a dermatologist 6 days after he had started to apply it. The eruption was successfully treated with a steroid ointment prescribed by the dermatologist. At this time, the patient noticed a new pruritic eruption on his right popliteal fossa, to which he also applied the steroid ointment. But, when the steroid ointment ran out, he started reapplying the Berybear cream. Over a period of 2 weeks, however, the eruption gradually got worse. The right leg showed diffuse erythema associated with severe tenderness and pruritus (Fig. 1a). Unilateral tender swelling of the leg indicated the possibility of deep vein thrombosis. Subsequent ultrasonography, laboratory data, and angiography excluded deep vein thrombosis, although stricture of the right femoral vein with bypass was detected (Fig. 1b). A skin biopsy showed numerous spongiotic vesicles with lymphoeosinophilic infiltration in the epidermis, and predominant numerous eosinophilic infiltrations intermingled with lymphocytes and histiocytes were present throughout the dermis and subcutaneous fat (Fig. 1c). Therefore, we diagnosed contact dermatitis and prescribed 20 mg of oral predonisolone and topical clobetasol propionate ointment. The symptoms resolved within 6 days. A 2-day closed patch test on the left upper armwith Berybear cream (as is) using Finn Chambers on Scanpor (Epitest, Tuusula, Finland) was performed. Pet. was used as negative control. Three healthy volunteers were also applied same materials. We assessed the reaction on D2, D3, and D7. International Contact Dermatitis ResearchGroup (ICDRG) (þþ) reaction was noted on D7, although negative on D2 and D3. Three volunteers did not show any positive reactions. A second closed patch testing using ingredients of Berybear cream, kindly provided by Daiichi Sankyo Co Ltd. and Ikeda Mohando Co Ltd. (Toyama, Japan) was performed. Among them, ICDRG (þþ) reaction was noted on D7 only to a pure product of hexyldecanol [INCI name; 2hexyldecanol is the CAS name; CAS number: CAS (2425-77-6)] (10% pet.). The reaction lasted until D21. The test for cetyl alcohol (6.5% pet.; concentration in Berybear cream) was negative, although it is structurally an isomer of hexyldecanol (Fig. 2). Three volunteers showednopositivereactions. The names of other ingredients have been withheld from this publication on instructions from the provider.

Subfascial lipoma in the temporal muscle that extended to under the zygomatic arch.

Immunostaining for human papilloma virus (K1H8; Dako, Glostrup, Denmark) was negative and MIB-1 (Dako) index was 12.0% (Fig. 1d). She was referred back to the previous dermatologist, but discontinued the visits after a 3-time trial of cryotherapy. The nodule started to grow with a bad smell 2 months prior. She was referred to us again. A nodule with dry necrosis and ulceration was seen (Fig. 1e,f). Dermoscopy showed a yellowish-white granular structure surrounded by reddish reticular structures (Fig. 1g). Biopsied histopathology was consistent with squamous cell carcinoma (SCC), and we resected the nodule with a 5-mm margin. Histopathologically, the epidermis had been replaced with exophytic growth of atypical keratinocytes with marked surface keratinization, cancer pearls, necrotic foci and frequent mitoses (Fig. 1h,i), compatible with verrucous carcinoma type SCC. Immunostaining against human papillomavirus (HPV) was negative, whereas MIB-1 index was increased to 25.2% (Fig. 1d,j). Adjacent to SCC, verrucous lesion were seen (Fig. 1k,l). The polymerase chain reaction (PCR) assay using 16 sets of genotype-specific primers was performed but no HPV genotype was identified. The patient has been tumor free for 8 months. This is the first case of a long-standing verrucous lesion ascertained histopathologically with subsequent development of SCC. Verruca vulgaris on the sole usually disappears within 2 years, and there are only a few reports of malignant transformation. However, the lesion was not examined at a premalignant stage in those studies. In our case, the lesion allegedly had not changed substantially until 2 months prior. It has been reported that HPV types 16 or 35 have a high-risk potential for malignant transformation via genital–digital spread. Although we performed multiple PCR analyses, we could not find any evidence of 14 highand 2 low-risk mucosal HPV genotypes. The patient might have been infected with other low-risk HPV genotypes, such as HPV type 2 which causes plantar wart, or the viral load decreased below the detection level or disappeared by the first visit 13 years later. The SCC in our case arose on the weight-bearing area and those receiving chronic mechanical stimulation may become aggressive. A study on organ-transplanted patients revealed a strong correlation between the existence of verrucokeratic lesions and the development of SCC. Long-term corticosteroid treatment for 10 years might have been involved in the development of SCC from the verrucous lesion in our case. Frequent biopsies or excision of a non-surgical treatment-resistant verrucous lesion is necessary, particularly in patients with an immunosuppressive status or with mechanical stimulation.

Percutaneous cardiopulmonary support as a possible risk factor for symmetrical peripheral gangrene of the toes

on his third toe of the left foot, but histopathological examination of the lesion revealed melanophages but no dysplastic cells. Then, we tried left groin dissection, and resection of the left pelvic and para-aortic lymph nodes as much as possible using the inguinal and pararectal approach. However, a part of the left pelvic and para-aortic lymph nodes, which were swollen, were not removed because of possible nerve injury and excess bleeding. Atypical epithelioid cells, which were positive for HMB-45, were found in the large part of the inguinal, pelvic and para-aortic lymph nodes (Fig. 1c,d). Hence, we considered that the primary lesion on the third toe of the left foot had spontaneously regressed, and malignant melanoma stage IV (pTxpN3M1a) was diagnosed. A combination chemotherapy including CDDP, DTIC, ACNU and tamoxifen (DAC-Tam) was not effective. Next, we performed percutaneous ethanol injection therapy (PEIT) with computed tomography (CT) guidance into the para-aortic nodes once a month in addition to i.d. injection of OK-432 once a week for 3 months (from August to October 2007). As a result, 34% reduction of the long axis of the para-aortic lymph node was achieved. After starting PEIT/OK-432 treatment, white macules appeared on the surgical site on his left inguinal region, thigh and face. Because the patient refused further treatment, no further antitumor therapy was conducted. Positron emission tomography/CT performed in November 2013 showed no accumulation (Fig. 1b). The patient has been healthy, having white macules on his face until February 2014. This case suggests that PEIT/OK-432 treatment after mass reduction surgery becomes an effective treatment for advanced melanoma with para-aortic lymph node metastasis. This finding is well compatible with a previous report which describes the rapid destruction of human melanoma by percutaneous injection of absolute ethanol, and another case report of advanced melanoma successfully treated with OK-432. The underlying mechanism of getting a long-term remission in this patient may be due to induction of anti-melanoma immunity by destruction of melanoma cells in the metastatic lymph nodes. Immunohistochemical study of the metastatic lymph node revealed marked infiltration of CD11band PD-1-positive cells (Fig. 1e–h) around the melanoma cells into the lymph node, supporting the high anti-melanoma immunity in this patient. Now, nivolumab, a new biologic which targets PD-1, is available for the treatment of melanoma, indicating that immunotherapies may improve survival outcomes in melanoma.

Partial response of angiosarcoma of the scalp to sorafenib: association with decreased expression of vascular endothelial growth factors and their receptors

The prognosis of angiosarcoma remains very poor, even with combined, multimodal therapy. We report a case with partial response of angiosarcoma of the scalp to sorafenib, which is a new oral, molecular, targeted, multiple‐kinase inhibitor. In addition, we confirmed, using immunohistochemistry, that sorafenib suppressed the expression of vascular endothelial growth factors and their receptors on the angiosarcoma tumour cells, and decreased cell numbers by inhibiting cellular proliferation.

Recalcitrant subungual verruca of a child successfully treated with combination use of traditional Japanese herbal medicines, shokenchuto and makyoyokukanto

action. Acta Derm Venereol 2010; 90: 18–22. 6 Piccinni MP. T cells in normal pregnancy and recurrent pregnancy loss. Reprod Biomed Online 2006; 13: 840– 844. 7 Challis JR, Lockwood CJ, Myatt L et al. Inflammation and pregnancy. Reprod Sci 2009; 16: 206–215. 8 Kus S, Candan I, Ince U, Uygur T. Eosinophilic pustular folliculitis (Ofuji’s disease) exacerbated with pregnancies. J Eur Acad Dermatol Venereol 2006; 20: 1347– 1348. 9 Georgala S, Korfitis C, Ioannidou D, Georgala C. Atypical Ofuji’s disease exacerbated with pregnancy. J Eur Acad Dermatol Venereol 2009; 23: 1094–1095.

Rapid detection of Bartonella henselae heat shock protein DNA by nested polymerase chain reaction from swollen lymph nodes of a patient with cat-scratch disease

Dear Editor, Diagnosis of cat-scratch disease by dermatologists is not always straightforward. Eruptions of the disease involve small scratched skin lesions, and are easily overlooked. The only useful clue to the diagnosis is the history of exposure to cats or occasionally dogs. The nested polymerase chain reaction (PCR) technique was recently developed to detect Bartonella DNA from the specimens of the patients, and fast and specific diagnosis has been available for cat-scratch disease. While the presence of antibodies to Bartonella henselae supports the diagnosis of cat scratch disease, the specificity of the serological test needs to be improved. Here, we report a case of catscratch disease in which B. henselae DNA was detected from a swollen lymph node by nested PCR. A 75-year-old man visited our clinic in September 2006 because of high fever and swollen right inguinal