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Ectopic endometriosis is a gynecologic disorder which is defined by the presence of endometrium tissue outside the uterus. Isolated breast involvement is a very uncommon manifestation of ectopic endometriosis. We present a 16-year-old girl with compliant of left breast pain, fever, and breast enlargement from 3 days ago. The ultrasound of left breast showed a well-defined heterogeneous mass lesion with thick turbid fluid components measuring 6 × 3.5 cm, consistent with abscess formation. The mass was surgically excised without complication. The histopathology reports confirmed the left breast endometriosis. The main purpose of this report is for physicians to consider this diagnosis when facing an unusual manifestation of endometriosis, especially in areas where the prevalence of this disease is rare.