Breast implant–associated anaplastic large-cell lymphoma (BIA-ALCL) is a rare disease, with 166 cases confirmed in the United States and 885 worldwide.1 Squamous cell carcinoma of the breast implant capsule is an even rarer malignancy associated with breast implants, with only eight cases reported previously in the English-language literature.2–8 A 46-year-old woman at 26 weeks’ gestation presented to our clinic with a 4-month history of pain and swelling of her right breast. Her history was significant for aesthetic breast augmentation with submuscular, smooth, round saline implants and two previous revisions for capsular contracture. Cytologic evaluation of a recurrent, complex periprosthetic fluid collection revealed abundant squamous cells, mostly enucleated, and no CD30-positive lymphocytes. An ultrasound-guided biopsy of a capsular mass was positive for squamous cell carcinoma. Modified radical mastectomy with en bloc excision of the implant and capsule was performed on the right breast with right sentinel lymph node biopsy. Final pathologic analysis of the right breast revealed an ill-defined, firm mass measuring 6 × 4 × 3 cm. This was determined to be well-differentiated squamous cell carcinoma arising from the medial breast implant capsule and invading the adjacent breast parenchyma and skeletal muscle (Fig. 1). The implant itself was confirmed to be an intact Mentor (Irvine, Calif.) smooth, round saline implant. There was an opaque, tan periprosthetic fluid collection with “pasty, white debris.” The capsule was diffusely studded with tan–white nodules and had extensive squamous metaplasia and atypia. The breast tissue was benign, showing only lactational changes. All sentinel and nonsentinel lymph nodes were negative for metastatic squamous cell carcinoma. The mass itself was negative for estrogen, progesterone, and HER2-neu receptors. The patient underwent induction of labor at 35 weeks’ gestation in order to expedite adjuvant chemotherapy and radiotherapy, which she tolerated well. She was in remission 12 months after initiation of adjuvant therapy.Fig. 1.: Excised right breast specimen, posterior view. Forceps are at 1 o’clock.Breast implant capsule–associated squamous cell carcinoma shares presenting symptoms with BIA-ALCL, namely, late-onset breast edema in the setting of breast implants present for 15 years or longer.3,4,9 The implants in the eight previously reported cases of breast implant capsule–associated squamous cell carcinoma were not limited to textured or silicone implants, in contrast to current data on BIA-ALCL.3,9 Our patient did have textured implants at one time, but she did not recall how long they were in place. The outcomes for breast implant capsule–associated squamous cell carcinoma seem to be worse than those for BIA-ALCL, with multiple patients having metastases reported within 2 years of diagnosis3,4,6 (Table 1). The published (albeit limited) experience with breast implant capsule–associated squamous cell carcinoma suggests that this is a much more aggressive pathology than BIA-ALCL, with more aggressive surgical management, as well as adjuvant therapy, necessary for disease management.3,4,6 Table 1. - Characteristics of Previously Reported Cases Characteristics Alfaro et al. 2 Buchannan et al. 3 Kitchen and Paletta 5 Olsen 6 Olsen 6 Satgunaseelen 7 Zhou et al. 4 Zommerlei 8 Age at diagnosis, yrs 68 65 52 56 81 58 46 58 Time most recent implants in place, yrs 17 35 15 18 >33 Unspecified; index operation 29 yr before presentation 8 yr; index operation 21 yr before presentation 15 yr; index operation >25 yr before presentation Reason for implants Reconstructive Aesthetic Aesthetic Aesthetic Reconstructive — Aesthetic Aesthetic Presenting symptoms Swelling Swelling, pain Swelling, pain Swelling, pain, erythema Swelling, pain Swelling, pain Swelling, hardening Swelling, pain, erythema Imaging — Periprosthetic fluid; ruptured implant — — Cystic mass on ultrasound — Periprosthetic fluid collection — Implant type and texture Saline Silicone (polyurethane foam-covered) Silicone Silicone, saline Silicone — Silicone Smooth silicone, saline, smooth silicone History of revisions and cause — — Ipsilateral capsular contracture; no revisions One revision for bilateral capsular contracture — Three revisions, details not specified Two revisions, details not specified Multiple, recurrent ipsilateral capsular contracture Implant integrity — Ruptured Intact Intact Intact — Intact Intact Treatment “Surgical intervention” Radical mastectomy, adjuvant radiation Modified radical mastectomy Mastectomy, SLNB, adjuvant chemotherapy and radiation Mastectomy, adjuvant chemotherapy and radiation Mastectomy Explantation, capsulectomy, mass excision, adjuvant radiation Mastectomy, SLNB Surgical findings — Periprosthetic milky fluid; posterior capsule adhered to muscle Periprosthetic “sebaceous” material, 6-cm mass adhered to capsule Thick, white periprosthetic fluid, mass adhered to posterior capsule 5-cm mass adhered to posterior capsule — — 5-cm fungating mass adhered to posterior capsule; gray, keratinous fluid Follow-up — 8 yr — ~1 yr >5 months — 1 yr — Outcome — Alive, disease-free — Locoregional metastasis, palliative radiation Distant metastasis, death — Diffuse metastasis, death — SLNB, sentinel lymph node biopsy. Our patient had transaxillary, periareolar, and inframammary incisions used in her previous augmentation and subsequent revisions, putting her at risk for ductal transection, implant colonization with biofilm-producing organisms, and subsequent chronic inflammation, which may have led to squamous metaplasia and subsequent dysplasia. There is no mention of incision type in the other case reports, but this information would be useful to document. Our patient’s most recent revision surgery was in 2016, at which time there was periprosthetic fluid and a mass on her capsule at the same site where her SCC ultimately developed. This was reported as benign on biopsy but no pathology report was available for our review. A unique factor in our patient’s case was her concurrent pregnancy. To our knowledge, this is the first report of a gravid woman with BICA-SCC. Given the paucity of data surrounding this pathology, we are unable to draw any conclusions regarding whether our patient’s pregnancy contributed to her disease development in any way. Breast implant capsule–associated squamous cell carcinoma is a rare but aggressive disease whose etiology remains unclear. Further study of this disease is needed to better classify risks, treatment, and outcomes. Plastic surgeons should remain vigilant in the follow-up of patients who have undergone placement of breast implants. A differential diagnosis of breast implant capsule–associated squamous cell carcinoma in patients presenting with late-onset breast edema, particularly with a history of previous capsular contracture or revision surgery, should be maintained. A similar approach to diagnosing BIA-ALCL should also be applied to diagnosing this disease: aspiration of any periprosthetic fluid collection for cytologic analysis and biopsy of any mass present.9 The surgeon should include the possibility of breast implant capsule–associated squamous cell carcinoma when discussing the possible risks of breast implant placement with patients before surgery. DISCLOSURE The authors have no financial interest to declare in relation to the content of this article.