Cutaneous polyarteritis nodosa: a report of a case associated with melioidosis (Burkholderia pseudomallei).

Abstract

A 22‐year‐old Thai woman was hospitalized with a 1‐month history of high‐grade fever and slowly progressing multiple erythematous painful nodules on both legs. A history of arthralgia in the knee and ankle joints was presented.She had been treated with acetaminophen and a non‐steroidal anti‐inflammatory drug (NSAID) from a provincial hospital before, but without improvement. She denied the histories of upper respiratory tract infection, jaundice, tuberculosis, and taking any drugs before skin eruption.On physical examination, she had a temperature of 40 °C, a pulse rate of 90 beats per minute, a respiratory rate of 22 breaths per minute, and a blood pressure of 110/60 mmHg. She had multiple 1–4 cm erythematous, oval, tender, nodules on the pretibial area of both legs. Some nodules also had fluctuation and hemorrhagic bullae ( Fig. 1). Other physical examinations were normal, except for positive splenic dullness on percussion.Erythematous, subcutaneous nodules with hemorrhagic bleb on both legsimageTest results from the following laboratory studies were normal; hematocrit, platelet, urinalysis, serum urea nitrogen, creatinine, electrolyte, amylase, liver function test, venereal disease research laboratory (VDRL) test, anti‐streptolysin O, anti‐nuclear antibody, and hepatitis B surface antigen and antibody. Tuberculin test was negative. The chest X‐ray film was normal. Hemoculture and fluid culture from bullae showed no growth of organisms.The leukocyte count was 15.1×109/L, with a differential cell count of neutrophils 73%, lymphocytes 19%, monocytes 7%, and eosinophils 1%. Melioid titer (by indirect hemagglutination test) was 1 : 2560. Ultrasound of the abdomen revealed splenomegaly with multiple, variously sized hypoechoic lesions in the spleen. The liver, gall bladder, pancreas, and kidneys appeared normal.A skin biopsy specimen from the inflammatory nodule on the leg showed perivascular and transmural neutrophilic and lymphocytic infiltration of medium‐sized arteries in the dermal‐subcutaneous junction and fibrinoid necrosis of the vessel walls ( Fig. 2 and 3).Transmural and perivascular neutrophilic and lymphocytic infiltration of medium‐sized artery in dermal–subcutaneous junction (hematoxylin and eosin; original magnification, ×100)imageBased on the physical examination, laboratory investigations, and histopathology, it was believed that cutaneous polyarteritis nodosa (CPAN), associated with splenic abscess from melioidosis, was the probable diagnosis. Initially, the patient was treated with rest and ibuprofen 1200 mg/day. Although her arthralgia improved slightly, she still had fever and persistent skin lesions. After the results of abdominal ultrasound and melioid titer were reported, we stopped ibuprofen and started treatment as melioidosis. We gave ceftazidime and trimethoprim–sulfamethoxazole. Her fever decreased to normal, and the skin lesions subsided altogether within 10 days. No new skin lesions appeared. The antibodies were then changed to doxycycline and trimethoprim–sulfamethoxazole in oral form and ultrasound of the spleen was followed up 2 weeks later. There was a decrease in size and number of the hypoechoic lesions in the splenic parenchyma. She was discharged after admission for 3 weeks. The patient remains clear of symptoms after 11 months.