LAUSR

We are pleased to learn that our article was one of the most cited in the history of the journal. We wrote it because of the rapid rise in Australia of patients with seroma-only BIA-ALCL disease who were being told they had cancer when this did not accord with the evidence [1]. The article’s hypothesis that not all patients with BIAALCL had an inevitable malignancy was founded on the observed epidemiology of BIA-ALCL. In summary, the rapid rise in diagnosis had mirrored the adve‘nt and increasing adoption of cytological testing for the disease. There was no reason to suppose that BIA-ALCL was not present with the same incidence in textured implant-related late seromas prior to the advent of cytological testing in 2008 as afterwards. Such implants were widely used in Australia for 16 years before the first case of BIA-ALCL was recognised. The median interval from implantation to diagnosis was 7–8 years, and cancer registry data showed no increase in the incidence of non-Hodgkin lymphoma in women in the period 2000–2013 [2]. The existence of spontaneous regression and spontaneous resolution is an explanation of what happened to the seroma patients who had undiagnosed BIA-ALCL prior to the onset of cytological testing to look for it—they got better, often without surgical intervention [3, 4]. The two case studies in our article were presented as supportive clinical evidence, consistent with the hypothesis. Furthermore, an important context was provided that the WHO 2016 classification of BIA-ALCL as a new lymphoma was and remains provisional and therefore, by definition, is uncertain [5]. This fact had been and continues to be largely ignored by both the academic and lay media. Lastly and most importantly, we felt uncomfortable with the label of malignant ‘lymphoma’ for the noninvasive presentation that achieved 100% cure with surgery [6], especially when no residual disease was necessarily identifiable at that surgery. Whilst such perspectives did not accord with the then established view, it was for these fundamental reasons that our article might have been considered innovative and relevant. The article received strong criticism in the form of subsequent published letters [7–10]. However, the correspondence focussed on the two case studies whilst failing to acknowledge and address the centrality of the epidemiological evidence to the hypothesis. Further, we were astonished to discover unpublished criticism that even included an allegation that the manuscript was fraudulent. This caused the withdrawal of an invitation to the primary author to present the paper at an international academic meeting. Thankfully, the allegation was easily disproved and the ethos of adherence to the scientific principle of open discussion prevailed. The invitation was graciously re-instated and the paper presented [11]. Since the publication of our article and consistent with its hypothesis, the number of cases of BIA-ALCL diagnosed and the proportion without invasive disease have continued to rise [12]. That spontaneous regression occurs & Daniel Fleming [email protected]