An SLE patient with prolactinoma and recurrent granulomatous mastitis successfully treated with hydroxychloroquine and bromocriptine

Abstract

Granulomatous mastitis (GM) is a rare benign mammary lesion in which autoimmunity and hyperprolactinemia are considered possible etiological factors. GM has a high frequency of relapse and may lead to chronic ulceration and fistula if not treated properly. Here we report a case of a 22-year-old systemic lupus erythematosus (SLE) patient with three years' disease duration, stable on prednisone and hydroxychloroquine, who was found to have prolactinoma and recurrent GM after she discontinued medication on her own accord. The patient subsequently recovered and remained free of GM relapse under treatment of prednisone, hydroxychloroquine and bromocriptine. Though autoimmune disorders and prolactinoma were reported in GM, a coexisting condition of SLE, prolactinoma, and granulomatous mastitis has rarely been observed in one patient. We suggest our case as an illustrative example of the complex interaction between autoimmunity, neuroendocrine dysfunction, and manifestations in the breast: Immunological disturbances in the background of SLE, coupled with elevated prolactin levels secondary to a prolactinoma, may have predisposed the patient to the development of GM. The mammary lesion recovered and maintained free of relapse under immunosuppressive and antiprolactinemic therapy.

Chloroquine analogues in drug discovery: new directions of uses, mechanisms of actions and toxic manifestations from malaria to multifarious diseases

“…Recently, prolactinoma and recurrent granulomatous mastitis in SLE patients have been successfully treated with hydroxychloroquine. 19 In patients with CLE, a combination of hydroxychloroquine and quinacrine is more appropriate as initiation therapy than hydroxychloroquine or chloroquine monotherapy and improves the Cutaneous Lupus Erythematosus Disease Area and Severity Index (CLASI) score and the response rate in non-responders. 20 , 21 Hydroxychloroquine is beneficial for patients with membranous lupus nephritis.…”

Temporal resolution of idiopathic granulomatous mastitis with resumption of bromocriptine therapy for prolactinoma

“…Non-surgical recommendations include pharmacologic (i.e., steroids, methotrexate, azathioprine, tuberculosis medications) or close observation [1,3,6,17] . A recent case report in a patient with systemic lupus erythematosus (SLE) and prolactinoma found that treatment of both conditions (immunosuppressive therapy for SLE and bromocriptine for prolactinoma) were necessary to maintain remission of IGM [18] . In the largest series to date, Pandey, et al reported that 23% of 49 IGM patients had recurrence after corticosteroid treatment [1] .…”

Assertive clinical practice in managing patients with idiopathic granulomatous mastitis: Review of literature

“…These changes IL [ 7 , [24] , [25] , [27] , [28] ]. As reported by Saydam et al the significant differences in IL-22 and −23 levels between GM patients and the control group were thought to contribute to autoimmunity in GM pathogenesis [ 28 ] in addition, it is been reported that rheumatoid factor (RF), ANA, and anti-dsDNA antibodies were positive in all patients with GM without other clinical manifestations suggestive of autoimmune [ [31] , [34] ].…”