Breast implant capsule-associated squamous cell carcinoma is exceedingly rare, with only eleven previously published cases. The present study systematically reviews the current literature and describes an additional case in a 56-year-old patient who had undergone previous breast implant augmentation with textured silicone implants 22 years prior. Systematic review of the literature demonstrated a scarcity of reported cases, yielding only eleven other incidences. Symptomatology for this patient involved pain, swelling, and erythema of the right breast prior to eventual surgery. Magnetic resonance imaging reporting extensive oedema and a large periprosthetic effusion with lobulated changes. The patient proceeded with bilateral capsulectomies and histology demonstrated moderately differentiated squamous cell carcinoma, areas of squamous metaplasia, and a chronic inflammatory cell infiltrate. Postoperatively, a positron-emission tomography scan showed no concerning uptake of fluorodeoxyglucose and no evidence of metastatic disease. The patient proceeded to a right-sided total mastectomy and axillary lymph node biopsy. Final histology demonstrated remnant well-differentiated squamous cell carcinoma, whilst five lymph nodes were negative of disease. The patient received postoperative radiation therapy. A clinical history of swelling and pain appears to be a common presentation for this condition. Aspirations of periprosthetic collections containing squamous cells should be considered concerning for neoplasm. The presence of squamous metaplasia within the specimen provides some credence for transformation to invasive carcinoma mediated through chronic inflammation. The presence of perineural invasion would be worth reporting in future cases as it may confer similar risk characteristics as in cutaneous squamous cell carcinoma. A finding of remnant carcinoma during completion mastectomy provides support for an aggressive approach to surgical resection. LEVEL OF EVIDENCE V: This journal requires that authors assign a level of evidence to each article. For a full description of these Evidence-Based Medicine ratings, please refer to the Table of Contents or the online Instructions to Authors www.springer.com/00266 .