Background: Silicone lymphadenopathy is a recognized complication of silicone implant rupture. It occurs when silicone droplets migrate from breast implants to lymph nodes, resulting in the formation of granulomas (known as siliconoma) and lymph node enlargement. The ipsilateral axillary lymph nodes are most commonly involved but it can also affect contralateral axillary, supraclavicular, internal mammary and mediastinal lymph nodes.Case presentation: A 60-year-old woman with a history of left breast cancer who had undergone modified radical mastectomy (MRM) followed by left breast reconstruction with implant (30 years ago) presented with right axillary lymph nodes enlargement. An excisional biopsy of the two larger lymph nodes was performed to rule out malignancy. Pathologic examination showed features of silicone lymphadenopathy. Further examination with Ultrasound and MRI confirmed breast implant rupture. Conclusion: Silicone lymphadenopathy following breast augmentation and reconstruction primarily affects the ipsilateral axillary nodes. Contralateral lymph node involvement is rare and may occur several years after breast cancer diagnosis and can be the first sign of breast implant rupture. Although, the need to exclude malignancy in such cases is of outmost importance, silicone lymphadenopathy should also be considered in the differential diagnosis.