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Pseudotumor of the Breast: An Unusual Presentation of Wegener's Granulomatosis

To the Editor: We report a rare case of systemic Wegener’s granulomatosis (WG) that presented initially as a breast mass. WG may affect almost any organ system, but typically has a predilection for the upper respiratory tract, lungs, and kidneys. It rarely affects breast tissue, but previous cases have been reported in which the presumed initial diagnosis was localized or metastatic breast carcinoma. In most cases, breast disease developed following the initial manifestations of systemic disease, as in this case. In the majority of these the breast mass typically occurred within weeks to months of the onset of systemic symptoms. In some cases, however, as in this case, the breast mass presented months to years after initial symptoms (1–5). The patient was a 48-year-old woman who had previously been fit and well. In 2001 she developed persistent coryzal symptoms (blocked and intermittent runny nose), chronic earache, reduced hearing in the left ear, and a sensation of numbness on the left side of her face running from her nose to her ear. She was seen by an ear, nose, and throat (ENT) surgeon in March 2002 and was found to have a left middle ear effusion. This was drained and a grommet was inserted. A biopsy of the nasopharynx was also performed, which was normal, and no specific cause was found for her symptoms. More than a year later she presented to the breast surgeons with a 2-month history of a single lump in her left breast. On examination, a firm, tender, 1 cm breast lump was found in the outer part of the left breast, the appearance of which was consistent with fat necrosis on both mammography and ultrasound scan. Fine-needle aspiration of the lump revealed findings consistent with a benign inflammatory lesion with both acute and chronic inflammatory cells, rather than fat necrosis as initially suggested. No malignant cells were seen. The patient was later reassured and discharged from the breast clinic. Three months later she re-presented with the initial lump having become erythematous and painful; it subsequently burst and began discharging pus. In addition, she was noted to have developed another separate lump medial to the existing one. Two courses of oral antibiotics did not resolve her symptoms and she was admitted to the hospital with a high fever, nausea, vomiting, anorexia, malaise, and arthralgia affecting her right knee and both ankles. She also experienced some epistaxis in the 10 weeks prior to her admission. Microbiologic cultures were negative and ultrasound scan performed at the time showed a large deep abscess which was ulcerating in the upper half of the left breast. The patient’s inflammatory markers were raised (erythrocyte sedimentation rate [ESR] 119 mm/hr, C-reactive protein [CRP] 272 mg/L) and she had a normochromic, normocytic anemia and thrombocytosis. She also had a raised creatinine level with normal urea. Her condition continued to deteriorate despite broad-spectrum intravenous antibiotics and intravenous fluids and her renal function worsened. A punch biopsy of the breast showed prominent leukocytosis with large numbers of inflammatory cells, including giant cells. The histology was reported as suggestive of either a form of primary vasculitis with giant cells or a primary inflammatory process with a secondary vasculitis. A rheumatology opinion was sought and a primary systemic vasculitis, probably WG, was suspected. Urine sediment was found to be active with red cell casts and protein. Her condition improved following transfer to the local renal unit, where she was treated with cyclophosphamide and steroids. Immunology confirmed the presence of classical antineutrophil cytoplasmic antibody (c-ANCA), with proteinase 3 antibody being raised at 9.0 μ g /ml ( N = 0–2). It seems that the breast symptoms preceded the onset of a severe systemic vasculitis involving the kidneys and occurred 3 years after the initial upper respiratory tract symptoms. Interestingly, despite a normal nasopharyngeal biopsy, her upper respiratory tract symptoms have also significantly improved with immunosuppression. In conclusion, WG may present as a pseudotumor in breast tissue and may herald the onset of more severe disease. It is an important condition to differentiate from