Kikuchi-Fujimoto disease and breast implants: is there a relationship?

Abstract

Histiocytic necrotizing lymphadenitis, also known as Kikuchi-Fujimoto disease (KFD) is a subacute, necrotizing non-suppurative lymphadenitis that characteristically affects young adults, mostly women of Asian origin, while is more rarely observed in Western countries. KFD usually presents with isolated cervical lymphadenopathies, while constitutional symptoms are absent to mild. KFD has classically a favorable prognosis, although some cases with a more aggressive clinical course have been reported. The pathogenesis of KFD remains controversial. Hence, neither an infectious nor an autoimmune etiology have been confirmed so far; particular HLA class II alleles have been linked to KFD. Here, we report two cases of KFD occurring in patients with a history of bilateral silicone breast implants, highlighting the challenge that KFD poses to the differential diagnosis, particularly with T-cell lymphomas. Case 1: A 40-year-old female with a history of bilateral breast augmentation with silicone implants for cosmetic reasons, developed an isolated left axillary lymphadenopathy five years after her breast surgery, which was diagnosed as ALK-negative anaplastic large cell lymphoma (ALCL) at another institution. The patient underwent removal of her breast implants (which were negative for lymphoma at histologic examination) and was treated with six cycles of cyclophosphamide, hydroxydaunorubicin, vincristine and prednisone (CHOP). Six years later, she developed a second isolated, painless right laterocervical lymphadenopathy, tender and elastic in consistency. The case was suspicious for relapse and the patient underwent excisional biopsy at our institution. At this time, a review of the original diagnostic slides was also performed. Case 2: The second patient is a 37-year-old female, with an unremarkable past medical history who presented with an isolated, painless right cervical lymphadenopathy five years after a bilateral breast augmentation with silicone implants. An excisional biopsy of the lymph node was performed. The lymph nodes of patient 1 and patient 2 were reviewed and a diagnosis of KFD was performed in both cases. In particular for patient 1, the original sample (i.e. originally diagnosed and treated as ALK-negative ALCL, was received for review) and the new sample (i.e. suspicious for relapse) revealed similar features of KFD. The patients received anti-inflammatory therapy as the only treatment and after a follow-up of 39 months and seven