To the best of our knowledge

To the best of our knowledge, glans penis is an extremely rare site that develops EPS. Roest and Ratnavel reported the case of a 44-year-old man with d-penicillamine-induced EPS on the glans penis with cystinuria. It presented as a single, isolated erythematous scarring plaque with a pale depressed center on the glans penis that developed after recurrent episodes of asymptomatic ulceration, without coexistent EPS at other sites. This finding was different from that in our patient. As mentioned above, d-penicillamine-induced degenerative changes of elastic fibers could be found in the skin, mucosa, and other visceral organs; therefore, it is reasonable that similar changes can occur in the glans penis to cause penile EPS. In conclusion, we present a case of EPS caused by long-term administration of d-penicillamine with the involvement of glans penis. The atypical presentation and rarity of the disease could cause delayed diagnoses or misdiagnoses. This disease is important, as it represents a degenerative process induced by d-penicillamine and probably represents only the tip of the iceberg because of the potential involvement of other nonlesional EZ Cap Reagent GG and visceral organs. Prompt recognition of these adverse cutaneous effects and discontinuation of the drug may be necessary to prevent the possible occurrence of systemic sequelae.
Introduction Dermatomyositis (DM) is an autoimmune disorder that causes proximal muscle weakness and manifests with several characteristic skins rashes, including heliotrope periorbital rash, Gottron’s papules, Shawl sign, V sign, and periungual telangiectasia. Diagnosis of DM relies on typical clinical manifestations, elevation of serum muscle enzyme levels, and a characteristic electromyographic (EMG) pattern. Periorbital edema is commonly seen in inflammatory myopathy, but generalized edema is an unusual presenting feature. To date, there are 14 reported cases of generalized edema secondary to adult DM. Cases of juvenile DM with anasarca were also reported. Such patients usually have a highly active disease with pronounced muscle weakness and dysphagia, which require aggressive treatment. We report a female patient who had acute edematous DM, with a relatively good initial response to oral dexamethasone. After a brief improvement, she experienced a sudden worsening of muscle weakness, with oropharyngeal dysphagia and dysarthria, which improved gradually in 2 months after intravenous pulse corticosteroid therapy.
Case Report A 44-year-old female without past history of renal or heart disease, presented with a 2 month history of erythematous maculopapular skin rash over the face, V-neck, back, and all extremities. She also experienced proximal muscle weakness in association with generalized swelling for 2 weeks prior to admission. The patient noted a body weight gain of 7 kg in 4 weeks (from 68 kg to 75 kg). Physical examination revealed Gottron’s papules on the dorsal hands (Figure 1), a periorbital heliotrope rash, and a generalized non-pitting edema, which was most pronounced over the face and upper limbs, especially the hands. A neurological examination showed decreased muscle power in her shoulder and pelvic girdles. Laboratory evaluations revealed a white blood cell count of 6700/mm, a hemoglobin level of 10.8 g/dL, a platelet count of 319,000/mm3, an elevated muscle enzyme creatine kinase of 992 IU/L, aspartate transaminase (AST) of 106 U/L, and alanine transaminase (ALT) of 50 U/L. Albumin was 2.8 g/dL, but there was no proteinuria on urinalysis. Renal and thyroid function test results were unremarkable. Circulating antinuclear antibodies were weakly positive (1:80), but anti-Jo-1 antibody, anti-Ro/La antibody, anti-ribonucleoprotein antibody, anti-Smith antibody, and anticardiolipin antibody were all negative. The chest radiography, electrocardiography, and sonography of the abdomen were unremarkable. Sonography of the bilateral upper extremities showed diffuse edema of subcutaneous and fascia layers (Figure 2). Histological examination of a skin lesion over the right postauricular area showed vacuolar interface dermatitis compatible with DM. The patient refused to undergo EMG. With the exclusion of other causes for limb edema, a diagnosis of DM with severe subcutaneous edema was made. She was then treated with oral dexamethasone 6 mg/day, which resulted in an improvement in muscle weakness and edema in 9 days, and markedly decreased muscle enzymes. A search for internal malignancy produced negative results. The patient was discharged on oral dexamethasone 6 mg/day.