Introduction: Breast implant-associated anaplastic large cell lymphoma (BIA-ALCL) is still a rare extralymphatic lymphoma. As of March 1, 2023, approximately 1,355 cases of BIA-ALCL have been reported worldwide. However, no such case has yet been described with pectoral implants in male patients. Most patients with BIA-ALCL present with nonspecific implant-associated symptoms such as late-onset seroma, swollen breasts, and deformation of implants. Case Presentation: Here, we describe BIA-ALCL in a 76-year-old male patient who presented with a late-onset seroma in order to raise awareness for BIA-ALCL also in men after esthetic chest surgery with silicone pectoral implants. The patient had undergone augmentation of the pectoralis muscle with implants for esthetic reasons 9 years before. First cytological specimens showed no malignancy. A repeated cytological assessment after 6 weeks from recurring seroma showed characteristic CD30+ T-cell clones. Surgery with complete bilateral capsulectomy and implant removal was performed. Due to the early-stage ALCL being limited only to the capsule and no evidence of systemic disease, adjuvant systemic treatment was not considered necessary. Conclusion: Any persisting late-onset seroma also in male patients with pectoral implants should raise suspicion of ALCL as differential diagnosis and should be assessed with cytological examination.

Established Facts

  • Breast implant-associated anaplastic large cell lymphoma (BIA-ALCL) is still a rare extralymphatic lymphoma.

  • BIA-ALCL patients often present with nonspecific implant-associated symptoms such as late-onset seroma, swollen breasts, and deformation of implants.

  • Any persisting late-onset seroma in patients with implants should be treated with cytological asservation.

Novel Insights

  • Breast implant-associated anaplastic large cell lymphoma (BIA-ALCL) can occur in pectoral implants.

  • Not only women but also men with late-onset seroma should raise suspicion of BIA-ALCL.

Breast implant-associated anaplastic large cell lymphoma (BIA-ALCL) is still a rare extralymphatic lymphoma. Not only women but also men undergo implant surgery due to various reasons such as transformation from male to female with breast augmentation, to correct pectus excavatum deformity, or cosmetic chest augmentation. Although BIA-ALCL has been described in transgender patients with breast augmentations as part of their transformation process, no such case has been reported in male patients with pectoral implants up to now. Therefore, to our knowledge, this is the first reported case of a male patient with pectoral implant to suffer from BIA-ALCL. Any persisting late-onset seroma also in male patients with pectoral implants should raise suspicion of ALCL as differential diagnosis and should be treated with cytological asservation.

A 76-year-old male patient presented at our center with spontaneous swelling of his right breast. He had undergone bilateral pectoralis muscle enhancement for esthetic reasons 9 years before. Subpectoral surgical placement of bilateral 190cc, textured (classified after EN ISO 14607:2018 as microtextured [10–50 μm] surface), silicone pectoral implants manufactured by POLYTECH, had been performed. Until then, he had not had any difficulties with the implants.

Two months earlier, he had presented in another clinic with the same symptoms, and a seroma puncture was performed, though without cytology. The patient presented without any history of trauma or systemic symptoms but claimed to have observed the swelling after direct sun exposure. Physical examination showed an asymmetric chest with a swollen right breast. Ultrasound showed periprosthetic fluid on the right side, although with both implants being intact. In both breasts, there was no evidence of solid masses. Axillary ultrasound implicated the existence of bilateral siliconomes. Extraction of 150 mL seroma on the right side was performed. Aspiration cytology showed lymphocytes, macrophages, granulocytes but no evidence of malignant cells (immunocytology performed for BerEp4, WT1, GATA3, CK7, mammaglobin, calretinin, claudin-4, CK7, CK 5/6, D2-40, S100, SOX10, melan-A, CD79a, PAX5, MiB1/Ki-67, CD10, CD3, CD20, CD7, CD4, CD30, and ALK). Microbiology showed no signs of yeast or bacterial growth. Regular consultancy after 2 weeks did not show any seroma, thus consultation only with recurrent swelling or pain was discussed with the patient. Re-consultation with recurrence of swelling happened 10 weeks later spontaneously without a history of trauma or further symptoms, again.

We performed another aspiration cytology of the recurrent seroma around the right implant. Cytology showed tumor cells being positive for CD4, CD30, and partially WT1, as well as a Ki-67 labeling index of >90%. A T-cell and B-cell clone analysis was performed showing clones within the T-cell line implicating the existence of a BIA-ALCL.

Staging with computer tomography (CT) of the thorax and abdomen showed no remaining periprosthetic seroma but unspecific, reactive mediastinal lymph nodes. No tumor mass was detected. Bilateral ultrasound of the breast and axillary showed no suspicious lesions. We discussed the case in the multidisciplinary tumor board. Bilateral en bloc removal was recommended. At surgery (5 months after first presentation), an en bloc capsulectomy was performed on both sides showing a thick capsule on the right and a thin-walled capsule on the left side.

Histology showed no more cellular signs of the cytologically detected ALCL resulting in stage IA disease (according to NCCN guidelines). Postoperative recovery of the patient was uncomplicated. The multidisciplinary tumor board recommended no further adjuvant treatment with stadium IA but clinical surveillance every 3–6 months for 2 years including sonography and CT scans. There was no further consultation by the patient over the last 6 months.

The case was reported to the responsible office for drugs and medical devices (to the Bundesinstitut für Arzneimittel und Medizinprodukte [BfArM]). In addition, contact was made with the production company and the implant was sent in for further examinations. In the final report, no defects in the product could be found. Furthermore, the production documents were checked. The product corresponded to the specifications in all respects at the time of the final inspection.

BIA-ALCL is still a rare extralymphatic lymphoma. Extranodal lymphomas with primary site of the breast comprise 0.5% of all breast malignancies and <1% of non-Hodgkin lymphomas [1]. Unlike predominantly B-cell-associated lymphomas in primary breast lymphomas without breast implants, implant-associated lymphomas are often of T-cell type [2]. The first case of anaplastic T-cell lymphoma associated with a breast implant was reported in 1997 by Keech et al. [3].

Since no globally working registry existed, the American Society of Plastic Surgeons (ASPS) has established a global network to share cases of breast implant-associated lymphomas, and as of March 1, 2023, they recognized approximately 1,355 cases of BIA-ALCL worldwide [4]. Recognized cases were seen in cis- and trans women with breast implants [5]. According to Hwang et al. [6], no specific type of implant, time from insertion, or integrity of the implant has been identified to be directly associated with the BIA-ALCL. There are various presented cases with male-to-female patients having undergone breast implantation for feminization procedures but not within cis-men due to esthetic chest transformation with specific pectoral implants. To our knowledge, there have been no reports of BIA-ALCL with pectoral implants in cis-men so far. This is the first case presenting with BIA-ALCL after chest surgery enhancement with pectoral implants in a male patient.

In 1988, pectoral implants were first developed to correct pectus excavatum deformity in men. By the early 1990s, these implants had found their way into cosmetic chest augmentation in male patients [7], which is the most common reason for pectoral implant surgery in men today [8].

The most common reported risk factor for developing a BIA-ALCL is a textured surface of the implant [9, 10]. Our patient also had textured pectoral implants. In 2017, Doren et al. [11] performed an US epidemiological study of BIA-ALCL and identified no patients with smooth implants. Many reported cases omit a description of the texture of implants; therefore, more research about risk factors being associated with BIA-ALCL is necessary.

As shown in our patient, the diagnosis of BIA-ALCL remains to be difficult due to variable clinical manifestations and absence of common awareness [10]. Therefore, it is important to document every case of BIA-ALCL in order to raise awareness and common knowledge. Most patients present with a late-onset seroma of the breast, which is defined as periprosthetic seroma arising more than 6 months after surgery without a history of trauma [12]. The US Food and Drug Administration (FDA) recommends aspiration of seroma and cytological and microbiological exploration in patients presenting with late-onset seroma [13]. There are different hypotheses about the origin of ALCL, most of them including chronic inflammation processes caused by the implant leading to emerging clonal lymphocytes due to chronical stimulation. This could also explain the predominance of ALCL in textured implants which tend to form a bacterial biofilm around them acting as possible basis for infections and therefore chronic lymphocyte stimulation [14, 15]. Continuous friction between capsule and implant surface could result in a chronic inflammation process, too [16].

Surgical treatment in early-stage disease includes total capsulectomy, removal of the implant, and any occurring mass with negative margins [17]. Due to most commonly unilateral involvement, there is no standard management of the contralateral breast. In our case, we performed a bilateral en bloc resection in order to minimize the risk of potential synchronous involvement [6] and to achieve symmetry. Patients presenting with higher stage ALCL should be considered for adjuvant chemotherapy [18].

Reconstruction of breasts following BIA-ALCL may be considered. Lamaris et al. [19] suggest immediate reconstruction being possible for ALCL confined to the capsule with smooth implants or autologous tissue. Patients with extensive disease should be considered for an interval reconstruction after 6–12 months and after having performed a tomography evaluation [19]. Higher stage disease requires adjuvant chemotherapy and local radiotherapy. The posttreatment surveillance for all stages includes physical examination every 3 to 6 months for 2 years and CT or PET-CT scans every 6 months for at least 2 years [18]. Longo et al. [20] recommended in their updated clinical recommendations being published in December 2022 a 5-year clinical follow-up for stage 1 and R0 resection. With a 5-year survival of more than 90%, prognosis of BIA-ALCL is quite good [18].

BIA-ALCL is still an extremely rare condition and the real incidence might be underestimated due to spontaneous settlement of late-onset seroma and the lack of histopathological surveillance. Thus, it is important to raise awareness and report every known case of breast or pectoral implant-associated ALCL. In Germany, all cases of ALCL are reported to the Federal Institute for Drugs and Medical Devices office (Bundesinstitut für Arzneimittel und Medizinprodukte [BfArM]).

Ethical approval from the Ethical Review Board was not applicable for this study. Ethical approval was not required for this study in accordance with local/national guidelines. Written informed consent was obtained from the patient for publication of this case report and any accompanying images.

Prof. Dr. Eike Burandt: advisory boards for Novartis and Daiichi Sankyo, and speaker’s honoria for AstraZeneca, Eisai, and NOGGO. All remaining authors have no conflicts of interest to declare.

No funding was received for this study.

Dr. Kerstin Riecke, Dr. Lisa Steinhilper, Dr. Charlotte von Bülow, and Prof. Dr. Isabell Witzel wrote the manuscript. All authors provided patient data for the evaluation. All authors read and approved the final manuscript.

Additional Information

Kerstin Riecke and Lisa Steinhilper contributed equally to this work.

All data generated or analyzed during this study are included in this article. Further inquiries can be directed to the corresponding author.

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