Kenji Kusatake

The First Human Case of Rickettsia tamurae Infection in Japan.

A case of Rickettsia tamurae infection in Japan is reported. A 76-year-old Japanese male had a tick bite which developed to local skin inflammation on his left leg. Anti-rickettsia antibodies were detected in his serum, and R. tamurae DNA was identified in his blood, the lesional skin, and the tick.

New zoonotic cases of Onchocerca dewittei japonica (Nematoda: Onchocercidae) in Honshu, Japan.

BackgroundZoonotic infections with Onchocerca species are uncommon, and to date only 25 clinical cases have been reported worldwide. In Japan, five previous zoonotic infections were concentrated in Oita, Kyushu (the southern island), with one previous case in Hiroshima in the western part of Honshu (the main island). The causative agent in Japan was identified as Onchocerca dewittei japonica Uni, Bain & Takaoka, 2001 from Japanese wild boars (Sus scrofa leucomystax Temminck, 1842). Here we report two infections caused by a female and male O. dewittei japonica, respectively, among residents of Hiroshima and Shimane Prefectures in the western part of Honshu.MethodsIn both cases, nodules were surgically removed. The parasites in nodules were identified on the basis of their histopathological characteristics. Identification was confirmed by sequencing the mitochondrial cytochrome c oxidase subunit 1 (cox1) gene from worms in the tissues used in the histological preparations.ResultsCase 1 was a 61-year-old woman from Hiroshima Prefecture who complained of a painful subcutaneous nodule on the back of her right hand. The causative agent was identified as a female O. dewittei japonica owing to transverse ridges on the cuticle and molecular analysis. Case 2 was a 78-year-old woman from Shimane Prefecture who had a painful nodule in the left temporal region. Histopathological characteristics and cox1 sequencing of the worm indicated that the causative agent was a male O. dewittei japonica.ConclusionsFor Cases 1 and 2, we diagnosed the causative agents as a female and male O. dewittei japonica, respectively. These findings indicate the spread of a zoonosis caused by O. dewittei japonica in the western part of Honshu, where wild boars have recently extended their habitats because of decreased annual snowfall, unused rice fields and a decline in the number of hunters in Japan. The O. dewittei japonica infection rate among wild boars was reported as 78% in Shimane Prefecture, in the western part of Honshu. Therefore, in the near future, zoonotic onchocercosis is likely to occur in Honshu as well as Kyushu, where wild boars, blackfly vectors and humans share the same habitat.

Fusion of single-photon emission computed tomography and computed tomography images of sentinel lymph nodes in extramammary Paget's disease of the scrotum.

We describe a new method for the fusion of single‐photon emission computed tomography (SPECT) and multidetector computed tomography (MDCT) images using an acrylic resin marker containing 99 m‐Tc phytate and viewing software (Fusion Viewer Version 1.0 for Windows). This method provided the fusion of SPECT and CT images in a short time and made it easy to identify the sentinel lymph nodes in the patient with extramammary Pagets disease of the scrotum.

Condylomata acuminata in the neovagina after male-to-female reassignment treated with CO2 laser and imiquimod

Condylomata acuminata are frequently occurring genital warts, but little is known about clinical features of the genital warts in transsexuals and their incidence. We report a case of condylomata acuminata arising on the neourethral meatus and the transplanted skin of the neovagina in a male-to-female transsexual, which was successfully treated with CO2 laser irradiation followed by topical application of imiquimod cream on the residual warts. This is a first report of using imiquimod cream for condylomata acuminata arising on the neovagina in a male-to-female transsexual.

Usefulness of sentinel lymph node biopsy for the detection of metastasis in the early stage of extramammary Paget’s disease

BackgroundInvasive cases of extramammary Paget’s disease have a poor prognosis, owing to its rapid progression and resistance to irradiation and chemotherapy. Morton et al. introduced a technique to identify sentinel lymph nodes by local injection of vital dyes and reported its benefit for managing the progress of malignant melanoma.ObjectivesSince the rate of lymph node metastasis of extramammary Paget’s disease is not uncommon (reported from 8.5% to 26%), this study tested the hypothesis that sentinel lymph node biopsy would be useful to detect lymph node metastasis in the clinically early stage of extramammary Paget’s disease.Materials & MethodsEighteen patients with primary extramammary Paget’s disease were enrolled in the study. The precise location of sentinel lymph nodes was visualized using either intraoperative patent blue dye injection or indocyanine green dye injection in combination with a hand-held gamma-detecting probe after a 99mTc phytate injection.ResultsOf the 18 patients, sentinel lymph node metastasis was identified in 2 (Cases 2 and 15) of the 2 patients with deep invasion and in 1 (Case 12) of the 7 patients with minimal dermal invasion, however, it was absent in 9 patients who had been categorized into the intraepidermal group.ConclusionThis study demonstrates that extramammary Paget’s disease possibly causes metastasis via regional lymphatic systems to which the lesion belongs, even in the early stages of no nodule or tumor formation, and that sentinel lymph node biopsy is useful to detect lymph node metastasis.

Stage IV malignant melanoma successfully treated with OK-432 and percutaneous ethanol injection therapy after mass reduction surgery

Dear Editor, The prognosis for patients with stage IV malignant melanoma has usually been poor because of a lack of effective chemotherapy. However, immunotherapy is known to enhance survival of stage IV malignant melanoma in association with appearance of skin depigmentation or leukoderma. Autoimmunity against melanocyte differentiating antigens has been considered to underlie the skin depigmentation/leukoderma and prolonged survival. A 70-year-old man was referred to our department complaining of a painful subcutaneous tumor in the left inguinal region in January 2007. Upon consultation, a black macule was seen on the nail bed. Gallium scintigraphy showed isotope accumulation in the left inguinal, left pelvic and para-

Wet Earwax Phenotype Determined by ABCC11 Polymorphism is a Highrisk Factor for Acne

Background: The type of earwax-wet or dry-is determined by a single nucleotide polymorphism (SNP)-538G>A (Gly180Arg)-in the ABCC gene (ABCC11), which encodes an ATP-binding cassette domain. Since axillary osmidrosis is closely related to earwax type, some dermatoses may be associated with earwax type. Objectives: We analyzed the association between the prevalence of a series of dermatoses and earwax phenotype. Further, we investigated ABCC11 localization in the skin. Methods: A total of 660 patients who visited Nopporo Dermatology Clinic in Hokkaido were enrolled in this study. The patients were interviewed to determine their earwax type-wet or dry-and a medical practitioner diagnosed dermatoses. The association between the prevalence of dermatoses and earwax type was analyzed statistically. ABCC11 localization in the skin was immunohistochemically evaluated using anti-ABCC11 antibody. Results: Wet earwax type was more prevalent in patients with acne than in those without acne and the odds ratio was 5.36. On the other hand, dry earwax was more prevalent in patients with atopic dermatitis than in those without it, and the odds ratio was 1.86, which was non-significant. In the immunohistochemical staining, ABCC11 was found to be strongly expressed in the sebaceous glands of wet earwax type skin specimen. Conclusion: Wet earwax phenotype is a high-risk factor for acne. Therefore, hyper-secretion of lipids from sebaceous glands elicited by ABCC11 may play an important role in the pathogenesis of acne.

Case of mucous membrane pemphigoid with immunoglobulin G antibodies to the beta 3 subunit of laminin‐332 showing clinically Stevens–Johnson syndrome‐like generalized blistering mucocutaneous lesions

laminin-332. The case was diagnosed as psoriatic erythroderma with autoimmune subepidermal bullous lesion of unknown etiology. Oral prednisolone (30 mg/day) with 0.12% betamethasone 17-valerate and maxacalcitol (25 lg/g) ointment suppressed new blister formation. However, during tapering of prednisolone (at 15 mg/day) new blister formation was observed. Addition of cyclosporin 100 mg daily with prednisolone was effective for both bullous and psoriatic lesions, and the skin lesions disappeared within 2 weeks. Prednisolone discontinued, and the patient was well controlled with cyclosporin 100 mg every other day without new blister formation. The usefulness of the combination therapy of systemic steroid and cyclosporin for pemphigus has been described. According to the Japanese guideline for the treatment of pemphigus, cyclosporin is graded in C1. To the best of our knowledge, this is the first case of psoriatic erythroderma with subepidermal bullae successfully treated with cyclosporin and prednisolone. Despite the presence of autoreactive antibodies in the patient’s serum, we could not identify the target molecule of the antibody. From the indirect immunofluorescence using 1 mol NaCl-split skin showing IgG reactivity with dermal sides of the split skin, differential diagnosis included epidermolysis bullosa acquisita, anti-laminin-332 pemphigoid and anti-p200 pemphigoid. Anti-p200 pemphigoid which turned out to be anti-laminin-c1 pemphigoid is known to be closely associated with psoriasis and almost one-third of 61 cases reported were those with psoriasis. Without positive findings of the specificity of the patient’s antibodies, no definite diagnosis of this case was established. The reasons that the patient’s sera did not react with the candidate molecules may include: (i) a low titer of autoantibody in the patient’s serum; (ii) a conformational change during the extraction of dermal substance in immune blot; or (iii) a novel pathogenic molecule in our case. Further analysis of the autoantigens would be required in terms of pathophysiology of autoimmune blistering disorders, which are occasionally observed in psoriasis.