Ketoconazole, an imidazole derivative known to inhibit cytochrome P450-dependent adrenal enzymes was given to a patient with a functioning adrenal rest tumor of the liver in preparation for surgery. The drug was administered in a stepwise manner for 42 days starting with 400 mg and reaching 1 g the last 4 weeks of the trial. Clear clinical improvement was evident early in the trial and was associated with evidence of amelioration of her hypercortisolism and striking changes in serum and urinary levels of steroid hormones and metabolites. Sex steroids in serum and urine fell dramatically from the first day to the end of the trial. Urinary 17-ketosteroid excretion fell from a basal average of 139 mg/24 h to near normal levels within a week of therapy; serum testosterone fell from a basal level of 2.4 to 0.18 ng/ml; serum 17 beta-estradiol fell likewise from 1096 to 150 pg/ml. In contrast, cortisol levels in serum and urine increased in the first 2 weeks of the trial and subsequently fell to values below the basal levels. Similarly, serum 17 alpha-OH-progesterone levels increased 63% above the basal levels by day 6 of the trial and declined afterwards. Nine months after successful tumor resection the patient is apparently cured as judged by steroid hormone levels and physical appearance. We conclude that ketoconazole was effective in blocking tumoral steroidogenesis which resulted in clinical benefit.

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Introduction Cystic neutrophilic granulomatous mastitis (CNGM) is a form of breast lobulocentric granulomatous inflammation characterized by cystic spaces of dissolved lipid, surrounded by neutrophils. Corynebacterium is frequently the etiological agent, specially C kroppenstedtii. It is often painful and can simulate malignancy by clinical and imaging features. Clinical Case A 36-year-old woman, three pregnancies with puerperal mastitis in all. Consults by increase in left breast volume, painful, 1-month evolution, with erythema and not quantified fever. With antibiotherapy, erythema resolved, but the mass persisted. Ultrasound showed a 28-mm right nodule and extensive left architecture distortion, hypoechogenic, poorly delimited, which compromised superior quadrants, both BIRADS 4. Bilateral core biopsies had typical findings of CNGM, with bacteria inside cysts, identified with HE, which were Gram positive. The cultures gave Corynebacterium, typified as tuberculostearicum by MALDI-TOF methodology, with antibiotic multisensitivity. After 6 weeks of evolution, the last two with ciprofloxacin, on the left the mass had decreased, with 3-cm periareolar collection. Right lesion fistulized on the biopsy path. Conclusions We present a case of bilateral cystic neutrophilic granulomatous mastitis with clinical and imaging features suspected of malignancy. After 6 weeks of evolution, the last two with ciprofloxacin therapy, she had reduction of volume of the larger lesion and fistulization contralateral. She would probably require in its management, in addition to surgery to resolve the fistula, use of lipophilic antibiotics and corticosteroids.