Hisashi Nomura

Unexpected recalcitrant course of drug-induced erythema multiforme-like eruption and interstitial pneumonia sequentially occurring after nivolumab therapy

Vemurafenib improves survival of melanoma patients. However, cutaneous side‐effects commonly occur in them. Nivolumab and ipilimumab are monoclonal antibodies against programmed death 1 and cytotoxic T‐lymphocyte‐associated antigen 4, both of which regulate excessive T‐cell activation. Although these agents induce antitumor immunity against melanoma, the modified immune condition may result in an unexpected adverse reaction which has not been observed previously. Herein, we report a case who manifested severe erythema multiforme‐like eruption with mucosal involvement associated with vemurafenib following nivolumab. The patient also subsequently suffered from ipilimumab‐induced interstitial pneumonia with refractory course. Such a case has never been reported. This case suggested that dermatologists should pay special attention to unexpected adverse events of these drugs, and carefully observe cutaneous and respiratory status of patients during the treatment of melanoma.

An elderly patient with chronic active Epstein–Barr virus infection with severe hydroa vacciniforme-like eruptions associated with αβT-cell proliferation

1 Marzano AV, Capsoni F, Berti E, Gasparini G, Bottelli S, Caputo R. Amicrobial pustular dermatosis of cutaneous folds associated with autoimmune disorders: a new entity? Dermatology 1996; 193: 88–93. 2 Antille C, Frei M, Sorg O et al. Amicrobial pustulosis of the folds associated with auto-immune disorders. A case report with an analysis of cytokine expression profile in skin lesions of cutaneous neutrophilic lupus. Dermatology 2008; 216: 324–329. 3 Lee HY, Pelivani N, Beltraminelli H, Hegyi I, Yawalkar N, Borradori L. Amicrobial pustulosis-like rash in a patient with Crohn’s disease under anti-TNF-alpha blocker. Dermatology 2011; 222: 304–310. 4 Pavlidakey P, Mills O, Bradley S, Casey J, Morgan MB. Neutrophilic dermatosis revisited: an initial presentation of lupus? J Am Acad Dermatol 2012; 67: e29–e35. 5 Lipsker D, Saurat JH. Neutrophilic cutaneous lupus erythematosus. At the edge between innate and acquired immunity? Dermatology 2008; 216: 283–286.

Blastic Plasmacytoid Dendritic Cell Neoplasm in a 7-year-old Girl with a Solitary Skin Lesion Mimicking Traumatic Purpura

© 2015 The Authors. doi: 10.2340/00015555-1903 Journal Compilation

GVHD-like erythroderma in the clinical course of thymoma-associated myasthenia gravis

Graft-versus-host disease (GVHD) occasionally occurs during transplantation or transfusion. It is rarely associated with thymoma and can present as a symptom of thymoma-associated multi-organ autoimmunity [1]. It has unfavourable prognosis due to complications, such as colitis or liver dysfunction [1-3]. Although, thymoma is a benign tumour that is radically resectable at an early stage, it is sometimes accompanied by autoimmune disorders and requires long-term management. Myasthenia gravis (MG) [...]

Coexistence of acquired perforating dermatosis and bullous pemphigoid: three cases.

Both acquired perforating dermatosis (APD) and bullous pemphigoid (BP) are associated with a wide variety of systemic and cutaneous disorders [1, 2]. However, coexistence of APD and BP has never been reported. Herein, we describe three patients with coexisting BP and APD. The patients had several common features, suggesting that this condition is a new and unique clinical presentation associated with BP.Patient 1 was a 65-year-old male who presented with a three-month history of pruritic skin lesions [...]

Cutaneous Mycobacterium massiliense infection after body piercing

A 19-year-old Japanese woman had her navel pierced for aesthetic purposes in a medical clinic. Aftercare by the practitioner was not provided, and she disinfected the surgical site herself every day. After two weeks, she developed a suppurative wound on the navel. The lymph nodes (LNs) in the groins became swollen one month later, and the LN in the left groin festered. She was admitted to our hospital and was started on empirical antibiotic treatment, which was ineffective. A computed tomography [...]

Case of Rapid Progression of Hemiatrophy on the Face: A New Clinical Entity?

A lot of diseases, including lupus profundus, morphea, lipodystrophy, and Parry-Romberg syndrome, may manifest progressive hemifacial atrophy. These diseases usually progress slowly and rapid progression of atrophy is extremely rare. We report a case of elderly-onset rapid progression of hemifacial atrophy only in three weeks. Our case did not meet variable differential diagnoses. We discuss the clinical character of the patient against the past of literature and suppose it may be a new clinical entity.

A patient with lupus miliaris disseminatus faciei treated successfully with a combination of oral metronidazole and topical tacrolimus

Dear Editor, Lupus miliaris disseminatus faciei (LMDF) is a rare granulomatous, inflammatory dermatosis of unknown etiology. It is characterized by reddish yellowish brown papules on the central face, particularly on and around the eyelids. There is no established therapy for LMDF due to lack of controlled study. Although many therapies, including tetracycline, oral steroids, dapsone, and topical tacrolimus are reported to be effective, there is no report of treatment with oral metronidazole in English literature. Herein, we report the first case of LMDF treated successfully with oral metronidazole. A 37-year-old woman visited our department with a 2-month history of almost asymptomatic eruption of the central face. She had tiny (1–3 mm), dome-shaped reddish yellow papular lesions without rash and teleangiectasia on the face including the eyelids, perioral area, forehead, and chin (Fig. 1a). Dermoscopy revealed apple-jelly nodule-like appearance (Fig. 1b). Laboratory examination, including angiotensin-converting enzyme and lysozyme, was within normal limits and the T-SPOT tuberculosis test was negative. The histopathology of papules on the right cheek revealed granulation tissue composed of central caseation necrosis surrounded by epitheloid cells (Fig. 1c). There were multinucleate giant cells of the Langhans type (Fig. 1d). The tissue was negative for Ziel-Neelsen stain. A diagnosis of LMDF was made. We had treated the patient with oral roxithromycin (150 mg twice daily) and topical tacrolimus for 1 month, however, the eruption showed no improvement. Oral metronidazole (250 mg twice daily) was then administered, while topical tacrolimus was maintained. Two weeks later, a prominent improvement was achieved. The eruptions almost disappeared with no scarring (Fig. 1e). The medication was maintained and no recurrence and side effects have occurred at 4-month follow-up. Metronidazole is a nitroimidazole antimicrobial drug. It is used both systemically and topically for many cutaneous disease, including rosacea, acne and perioral dermatitis. However, the use of oral metronidazole for treatment of LMDF has never been reported in English literature. LMDF is a distinctive disease entity of a debatable pathophysiology, and a variety of treatments have been tried and reported with variable outcomes. There are some reports of the treatment for rosacea, one of similar conditions of LMDF, by oral metronidazole. Pye et al. conducted a double-blind trial in 29 patients with rosacea and showed that metronidazole was therapeutically superior to a placebo after 6 weeks’ treatment. No other patients presented side effects during the trial. Saihan et al. conducted a randomized double-blind trial in forty patients and showed the same effectiveness of oral metronidazole to rosacea as that of oral oxytetracycline after 6 and 12 weeks’ treatment. No side effects were reported. Although these trials provided only limited data, oral metronidazole might be a choice of treatment for rosacea and its relative conditions including LMDF. We treated the patient with oral metronidazole in combination with topical tacrolimus, therefore we can not rule out the possibility that topical tacrolimus is also effective. However, for 1 month before the introduction of metronidazole, we also had used topical tacrolimus, but the eruption showed no improvement. Therefore, we believe the oral metronidazole was most effective in our patient. Figure 1. (a) Clinical features on the first visit. (b) Dermoscopic images. Apple-jelly nodule-like appearance. (c) Granulation tissue with central caseation necrosis. (d) Langhans cells. (e) Clinical features after the treatment.

Case of lamotrigine-induced drug adverse reaction under tocilizumab treatment with clinical and virological features of drug-induced hypersensitivity syndrome

The pathological mechanisms and immunological kinetics of drug‐induced hypersensitivity syndrome (DIHS), including the relevance of interleukin (IL)‐6, remain unclear. We report a case of drug adverse reaction that does not fulfill the diagnostic criteria of DIHS but mimics its characteristic features. Because the patient was under anti‐IL‐6 therapy at the onset, some symptoms typically seen in DIHS were absent, such as fever and leukocyte count abnormalities. However, the characteristic features of DIHS were clearly observed in the subsequent course, including the repeated recurrence of skin rash, prolonged liver dysfunction and reactivation of herpes viruses. This case suggested that IL‐6 role at the onset is not a main factor to determine the subsequent pathomechanism of DIHS and attention should be paid to the preceding therapy for achieving accurate diagnosis.

Bullous pemphigoid in infancy with spontaneous remission.

A boy, aged three months and nine days, was noted to have small papules on his limbs seven days before receiving bacillus Calmette-Guerin vaccine, which was followed, nine days later, by diphtheria-pertussis-tetanus (DPT) vaccine. Two days later, abdominal erythema appeared. About three weeks after the DPT shot (at the age of four months and 15 days), tense blisters emerged on the face and extremities, and spread to the body. His mother had faced no similar symptoms during pregnancy. The patient [...]