Kohei Nojima

Drug-induced hypersensitivity syndrome by ethambutol: A case report

induced by levetiracetam. Seizure 2012; 21: 823–825. 3 Naranjo CA, Busto U, Sellers EM et al. A method for estimating the probability of adverse drug reactions. Clin Pharmacol Ther 1981; 30: 239–245. 4 Bunnell K, Pucci F. Levetiracetam-induced neutropenia following traumatic brain injury. Brain Inj 2015; 29(1): 115–117. 5 Bonastre MT, Munoz SP, Boza FM, Padro JG. Neutropenia secondary to exposure to levetiracetam. Tumori 2015; 101: e145–e146.

Case of tumoral melanosis with a massive infiltration of CD163+ and CD68+ macrophages

chronic hand eczema refractory to topical corticosteroids. Also, the earlier studies have suggested that alitretinoin could be a meaningful treatment option for PPP. However, its efficacy was not demonstrated in a recent controlled study. Acrodermatitis continua of Hallopeau and PPP are closely related and nearly indistinguishable clinically. However, compared with PPP, ACH tends to not respond to conventional treatment of psoriasis. Effective therapeutic alternatives also need to be presented. Herein, we experienced a case of refractory ACH successfully treated using oral alitretinoin and noticed dramatic improvement of nail abnormality as well as skin lesions. Although this is a single-case study, we think our experience is worthy of report. In addition, further studies will be needed. We suggest that oral alitretinoin is a reasonable and reliable therapeutic option for recalcitrant ACH in case of failure of other treatment agents.

Selective Plasma Exchange for the Removal of Pemphigus Autoantibodies, Fibrinogen, and Factor XIII in Pemphigus Vulgaris

Pemphigus vulgaris is a serious autoimmune skin disorder associated with desmoglein 1 and 3. Selective plasma exchange (SePE) for pemphigus vulgaris remains unknown. We investigated the removal characteristics of pemphigus autoantibodies, immunoglobulins, and fibrinogen in three cases. When the mean processed volume for SePE was 1.2 plasma volumes, the mean percent reduction was 50.7% for desmoglein 1, 48.9% for desmoglein 3, 50.3% for IgG, 29.8% for IgA, 1.9% for IgM, and 17.6% for fibrinogen. In one case, the percent reduction after four sessions of SePE within eight days was 87.0% for desmoglein 1, 85.1% for desmoglein 3, 76.6% for IgG, 53.5% for IgA, 7.9% for IgM, 41.6% for fibrinogen, and 31.4% for factor XIII. SePE can effectively remove pemphigus autoantibodies and retain coagulation factors, e.g. factor XIII and fibrinogen. In severe cases, SePE can be useful and safe for induction therapy.

Ulcerated giant pilomatricoma with appearance of cutaneous malignancy on positron emission tomography/computed tomography

had been pointed out to his parents at birth; however, the details of the disease were not explained. There was no family history of albinism or a hemorrhagic tendency. He was diagnosed with Crohn’s disease at 24 years old. He had worked as a clerk, but had rarely used sun block. He noticed a small tumor of the right cheek at 41 years old. After 2 years, the tumor rapidly increased and marked hemorrhage was noted, and he visited our department. On physical examination, OCA, nystagmus and visual disorder were observed. There was a 45 mm 9 45 mm tumor on the right cheek (Fig. 1a), and the biopsy findings suggested SCC (Fig. 1b,c). Computed tomography revealed that the tumor was adjacent to the masticatory muscle (Fig. 1d). Two courses of chemotherapy with cisplatin and fluorouracil were conducted (Fig. 1e), and tumorectomy and reconstruction using a free forearm flap was performed. Although the fibrinogen level, prothrombin time, partial thromboplastin time and platelet count were normal, the bleeding time was markedly prolonged (12.5 min), and it was difficult to stop bleeding during the surgery. Therefore, detailed platelet aggregation analysis was performed. Arachidonic acidand thrombin receptor agonist peptide-induced platelet aggregation was reduced, whereas adenosine diphosphateand collagen-induced aggregation was not impaired. This indicated impaired secondary platelet aggregation, which is consistent with the features of HPS. We genetically screened for mutations of his HPS1 by the simultaneous single-strand conformation polymorphism method, and detected a pathological mutation (c.2003T>C, p.L668P, homozygote), which has been reported. Based on the results of sequencing analysis with the platelet aggregation test, a diagnosis of HPS1 was made. After the surgery, strict guidance for sunlight avoidance was provided to prevent AK and SCC development. Multiple AK lesions have been treated with imiquimod. There has been no local relapse or distant metastasis during the postoperative follow up of 3 years and 10 months (Fig. 1f), and activity of an intestinal lesion, which had been diagnosed as Crohn’s disease, remains stable and there is no pulmonary lesion. Although the accurate incidence is unclear due to the small number of patients, OCA is sometimes complicated by AK and SCC. Our patient was diagnosed with OCA at birth, however, the explanation of the disease given to his parents had been insufficient, resulting in the development of multiple SCC and AK. Thus, this case illustrates that guidance towards sun protection for OCA patients is important. In addition, when we identify an OCA case, we should carefully assess the patient’s bleeding tendency and perform genetic analysis for early diagnosis. Consequently, we can predict or treat life-threatening manifestations in addition to prevent SCC development.

Phakomatosis pigmentovascularis type IIb: A case with Klippel–Trenáunay syndrome and extensive dermal melanocytosis as nevus of Ota, nevus of Ito and ectopic Mongolian spots

Dear Editor, Phakomatosis pigmentovascularis (PPV) has several variants due to different combinations of pigmentary lesions and vascular lesions. PPV type IIb is characterized by capillary malformation and dermal melanocytosis with or without nevus anemicus. Here, we present a case of PPV type IIb in association with Klippel–Tr enaunay syndrome. Our case is rare in terms of its extensive dermal melanocytosis. A 53-year-old man presented with a diffuse bluish gray pigmentation on his left cheek, right back and shoulder, and diffuse red-purple aggregated patches on his upper and lower limbs (Fig. 1a,b). There was no familial history of PPV or other

Dermoscopy of pigmented papillated Bowen disease: A report of two cases

(g) (h) Figure 1. (a) A 5 mm 9 3 mm brown keratotic plaque behind the left ear of the patient (case 1). (b) Dermoscopic findings showing whitish scaly fissures and bluegray to brown ridges surrounded by irregularly shaded brown areas. White arrows indicate fissures and a white arrowhead indicates ridges (case 1). (c) Histopathology showing a hyperkeratotic lesion with papillomatosis (case 1) (hematoxylin–eosin [HE], original magnification 920). (d) Atypical keratinocytes were hyperchromatic (case 1) (HE, 9400). (e) A 5 mm 9 3 mm dark brown keratotic nodule on the left knee of the patient (case 2). (f) Dermoscopic findings showing a blue-gray to brown cobblestone appearance with dark brown or whitish fissures and a continuing brown structureless area. White arrows indicate fissures and white arrowheads indicate ridges (case 2). (g) Histopathology showing hyperkeratosis, parakeratosis and papillary upward projections of the epidermis (case 2) (HE, 920). (h) Atypical keratinocytes were seen in the entire epidermis (case 2) (HE, 9200).

Amicrobial pustulosis-like rash associated with systemic lupus erythematosus

1 Yokogawa N, Tanikawa A, Amagai M et al. Response to hydrochloroquine in Japanese patients with lupus-related skin disease using the cutaneous lupus erythematosus disease area and severity index (CLASI). Mod Rheumatol 2013; 23: 318–322. 2 Ikeda T, Kanazawa N, Furukawa F. Hydroxychloroquine administration for Japanese lupus erythematosus in Wakayama: a pilot study. J Dermatol 2012; 39: 531–535. 3 Momose Y, Arai S, Eto H, Kishimoto M, Okada M. Experience with the use of hydroxychloroquine for the treatment of lupus erythematosus. J Dermatol 2013; 40: 94–97. 4 Wimmershoff MB, Hohenleutner U, Landthaler M. Discoid lupus erythematosus and lupus profundus in childhood: a report of two cases. Pediatr Dermatol 2003; 20: 140–145.

Well‐differentiated syringomatous carcinoma with solid carcinoma‐like features

1 Botet MV, S anchez JL. Vesiculation of focal acantholytic dyskeratosis in acral lentiginous malignant melanoma. J Dermatol Surg Oncol 1979; 5: 798–800. 2 Kartono F, Shitabata PK, Magro CM, Rayhan D. Discohesive malignant melanoma simulating a bullous dermatoses. J Cutan Pathol 2009; 36: 274–279. 3 Aneiros-Fern andez J, Arias-Santiago S, Diaz-Recuero JL, O’valle Ravassa F, Nogales Fern andez F, Aneiros Cachaza J. Acantholyticlike malignant melanoma: an unusual morphologic variant. Am J Dermatopathol 2010; 32: 364–366. 4 Vogt T, Brunnberg S, Hohenleutner U, Landthaler M. Bullous malignant melanoma: an unusual differential diagnosis of a hemorrhagic friction blister. Dermatol Surg 2003; 29: 102–104.

Case of subungual tumoral melanosis: The detection of melanoma cells and dermoscopic features

Dear Editor, Tumoral melanosis is a rare event, which substitutes melanin-laden macrophages (melanophages) for most melanoma cells. Although dermoscopic features of pigmented epithelioid melanocytoma and tumoral melanosis share black and blue homogenous areas, black streaks and crystalline structures may lack in tumoral melanosis. No detailed presentation of the dermoscopic features of subungual tumoral melanosis has been published to date. Here, we present a case of subungual tumoral melanosis. We emphasize the crucial importance of detecting residual melanoma cells using dermoscopy for the diagnosis of tumoral melanosis. A 67-year-old woman presented with a black tumor under the nail plate of her right thumb. The patient had initially recognized black streaks at the nail 5 years before. Those black streaks had gradually enlarged 2 years before. Physical examination revealed a 5 mm 9 10 mm black tumor under the nail plate of her right thumb. The nail plate was partially destructed and dented. A slight pigment macule was recognized at the lateral and distal periungual skin (Fig. 1a). Dermoscopy revealed a blue whitish veil on black homogenous areas with surface scales. A slight bleeding was found at the lateral portion of the black tumor (Fig. 1b). At the distal periungual skin, a parallel ridge pattern was found (Fig. 1c). The diagnosis of subungual melanoma was suspected. Her right thumb was amputated and a sentinel lymph node biopsy was performed due to the potential presence of melanoma cells in the black tumor mass. Histopathological examination revealed a massive infiltrate of melanophages under a large cleft of the

Pigmented Bowen disease: A challenging dermoscopic feature due to a traumatic disfigurement

Dear Editor, The characteristic dermoscopic features of Bowen disease have been reported. However, variations of the dermoscopic features of Bowen disease have not been fully elucidated to date. The dermoscopic features at the foot should be cautiously evaluated due to potential modifications due to mechanical stimuli such as trauma and irritation. Here, we present the dermoscopic features of Bowen disease at the foot with traumatic disfigurement. A 72-year-old man presented with a nodule with erosion on his foot. The patient had bruised the lateral malleolus of his foot 2 months before, and a nodule had developed at the bruise. Physical examination revealed an elevated 6 mm 9 6 mm dusky-red nodule with an erosion in the center on his lateral malleolus (Fig. 1a). Dermoscopy revealed glomerular vessels arranged annularly on a light-brown structureless area (Fig. 1b), and glomerular vessels were clearly seen at the edge of the erosion. At the base of the erosion, a pink homogenous area with slight whitish areas was seen (Fig. 1c). Brown structureless areas with a whitish veil were found at the periphery (Fig. 1d). Histopathological examination at the erosion of the tumor revealed the lack of a cornified layer (Fig. 1e). Elongations of rete ridges of atypical cells were prominent. Blood vessels ascended between the elongated tumor nests (Fig. 1f), and numerous mitoses were found (Fig. 1g). Histopathological examination at the periphery of the tumor revealed acanthosis with atypical cells (Fig. 1h) and melanocytes were scattered in the epidermis (Fig. 1i). Immunohistochemistry showed positive reactions for p63, p40, cytokeratin (CK)17 and AE1/3, and negative reactions for S100, adipophilin, p16 and CK1. Immunohistochemistry for a-smooth muscle actin and CD34 showed that blood vessels ascended and were partially dilated in the ascending dermis between the elongated tumor nests (Fig. 1j). Immunohistochemistry for Melan-A showed scattered melanocytes in the whole epidermis (Fig. 1k). Fontana–Masson staining showed melanin deposition both at the basal layer and in the spinous layer (Fig. 1l). From those clinical and histopathological findings, the diagnosis of pigmented Bowen disease was made. An additional resection of the tumor with a 5-mm surgical margin was performed, and there has been no relapse in the 5 months since then. The dermoscopic features of pigmented Bowen disease are characterized by both vascular structures and epidermal structures. However, those dermoscopic features may manifest atypical findings depending on several factors such as the location and mechanical stimuli. Mechanical stimuli, which results in traumatic disfigurement, may modify dermoscopic features as an enhancement of vascular structures in basal cell carcinomas (BCC). As a differential diagnosis, pyogenic granuloma, eccrine poroma, BCC, nodular melanoma and Bowen