Department of Medicine and Science of Ageing, University G. DAnnunzio, Via dei Vestini, 66100 Chieti, Italy
Correspondence to:
F. Di Stefano, Internal Medicine, Presidio Ospedaliero G. Bernabeo, C. da S. Liberata, 66026 Ortona (Chieti), Italy. E-mail: distefa.fabio{at}tiscali.it
SIR, Urticarial vasculitis (UV), which is characterized clinically by urticarial lesions that last for more than 24 h and resolve with purpura or hyperpigmentation, and histologically by leucocytoclastic vasculitis [1], has been described as a rare association with visceral and haematological malignancy [27]. UV has also been described in a few patients as developing reproducibly after exercise [8].
We report a patient who developed UV after exercise, who was incidentally diagnosed as having a cystic teratoma, and who did not suffer from the exercise-induced UV after the tumour had been surgically resected.
A 42-yr-old man presented with a 1-month history of recurring erythematous wheals over the lower extremities which appeared a few hours after physical exercise (on every occasion after his weekly jogging). Each lesion persisted for more than 24 h but disappeared after less than 3 days, leaving the area with slight pigmentation. He was examined during the eruption and blood was taken for investigation. He had a normal haemoglobin concentration and total and differential white blood cell counts. The erythrocyte sedimentation rate was 29 mm/h (normally <20 mm/h). C-reactive protein concentration, plasma protein electrophoresis, immunoglobulins, complement components, liver function tests, urea and creatinine, urinalysis, plasma sodium, potassium, glucose and thyroid function were within normal ranges. Screening for hepatitis B and C virus infection was negative. Antinuclear antibodies and cryoglobulins were not detected. Skin biopsy of an urticarial lesion demonstrated leucocytoclastic vasculitis. After complete resolution (within 3 days and without any therapy), challenge by physical exercise was performed to determine whether exercise could reproduce the urticarial lesions. Exercise challenge was performed on a cycle ergometer for a total of 6 min at room temperature. The workload was increased in a stepwise manner until 85% of the subject's predicted value of maximal heart rate was reached. Within 56 h after exercise challenge, urticarial lesions developed on the patient's legs and started to disappear at 72 h, leaving slight pigmentation. Skin biopsy was carried out at 24 h, and histology was again indicative of leucocytoclastic vasculitis. The investigations were completed with abdominal ultrasound, which was normal, and a chest X-ray, which showed a tiny widening of the superior mediastinum. Therefore, the patient underwent a chest computed tomography scan, which showed a cystic mass in the anterior superior mediastinum. A chest MRI scan revealed that this mass was very well demarcated and had the imaging features of a cystic teratoma (Fig. 1). The mass was surgically resected, and histology of the specimen confirmed the diagnosis of cystic teratoma. Six months after the operation, the patient restarted his weekly jogging and told us that he did not suffer the urticarial lesions after physical exercise. He repeated the exercise challenge, which did not provoke any cutaneous lesions.
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The patient gave informed consent to the investigations that led to the diagnosis reported here, and written permission was given for this case to be reported.
The authors have declared no conflicts of interest.
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