Idiopathic granulomatous mastitis with erythema nodosum and polyarthritis

Alungal, Jemshad; Abdulla, Mansoor C; Narayan, Radhika

doi:10.4081/reumatismo.2016.844

Cited by 20 publications

(13 citation statements)

References 7 publications

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“…Although cases of IGM complicated solely by EN are very rarely reported, to our knowledge, only ten other cases of IGM complicated by both arthritis and EN have been previously reported. All the patients received immunosuppressive treatment (9,10). It should be considered that IGM and EN may have similar autoimmune mechanisms during their development.…”

Section: Discussionmentioning

confidence: 99%

Coexistence of idiopathic granulomatous mastitis and erythemanodosum: successful treatment with corticosteroids

Akın¹,

Karabacak²,

Esendağlı³

et al. 2017

Turk J Med Sci

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Background/aim: Idiopathic granulomatous mastitis (IGM) is a rare, chronic inflammatory disease of the breast. Erythema nodosum (EN) is a rare extramammary manifestation of IGM. The purpose of this study is to determine the clinical and demographic characteristics of 11 IGM and EN patients and to evaluate the efficacy of methylprednisolone treatment. Materials and methods:In our series, ten patients had EN bilaterally, whereas one patient had a lesion of the right pretibial area. The mean age of the patients was 35.5 years (range: 29-45 years). IGM and EN were diagnosed by the necessary serological, microbiological, radiological, and histopathological examination. After diagnosis, methylprednisolone was started in the first week at 0.8 mg/kg daily for treatment. The weekly dose was tapered to 0.1 mg/kg daily over 8 weeks. Results:We started with the treatment of methylprednisolone, and in all our cases the initial response was excellent. In 2 weeks the IGM symptoms had markedly declined, while signs of EN disappeared completely. Patients were followed for an average of 60 months after treatment. None of the 11 patients had recurrence. Conclusion:We herein report a rare series considering IGM cases complicated by EN. Few such cases have been reported in the literature. We advocate for an initial trial of methylprednisolone treatment, which proved to be very successful in our patients.

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Section: Discussionmentioning

confidence: 99%

Coexistence of idiopathic granulomatous mastitis and erythemanodosum: successful treatment with corticosteroids

Akın¹,

Karabacak²,

Esendağlı³

et al. 2017

Turk J Med Sci

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“…The EN is a rare systemic symptom of IGM, first described by Adams et al in 1987 8 . The EN and IGM association is rare, reported with case reports and series 9‐15 . Additionally, to the best of our knowledge, it is the only systemic and extra‐breast finding of IGM.…”

Section: Introductionmentioning

confidence: 96%

“…8 The EN and IGM association is rare, reported with case reports and series. [9][10][11][12][13][14][15] Additionally, to the best of our knowledge, it is the only systemic and extra-breast finding of IGM. However, no studies were reported before elaborating the significance of both entities' coexistence on the severity of IGM and response to conventional treatment.…”

Section: Introductionmentioning

confidence: 99%

Idiopathic granulomatous mastitis with erythema nodosum: Is it a variant of clinical presentation indicating treatment resistance? A retrospective cohort study

2020

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Idiopathic granulomatous mastitis (IGM) rarely occurs with erythema nodosum (EN) as a systemic finding. However, the impact of their coexistence on disease severity and response to steroids has not been investigated yet. Patients diagnosed with IGM between September 2014 and October 2018 were divided into two groups according to the presence or absence of EN during the first admission retrospectively. The IGM was more severe in patients with EN as it was presented more often as bilateral and diffuse involvement of the breast. Findings of mastitis did not resolve with steroids in 50% of this group. Repetitive excisions and mastectomy with reconstructions were required to control the disease. Coexistence of EN and IGM was found to be related to bilateral and aggressive involvement, which could be associated with insufficient response to steroids. Associated patients should be informed in terms of the aggressive course, and surgery can be highlighted as a first-line treatment.

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“…Recently, autoimmunity and immune dysregulation have been emphasized, but studies on this subject are limited. Some patients with IGM respond well to immunosuppressive drugs such as steroids and methotrexates; the presence of extramammary findings likes erythema nodosum or arthritis in some patients and some recent studies, although limited, suggest that more emphasis should be given to autoimmunity and immune dysregulation in IGM [3][4][5][6][7][8][9][10][11][12][13]. The aim of our study was to investigate the number of T and B lymphocytes and changes in NK and NKT cells in IGM patients.…”

Section: Discussionmentioning

confidence: 99%

“…Remission and relapse of IGM in some patients with rheumatologic findings, such as arthritis and/or erythema nodule, suggest autoimmunity and immune dysregulation [5][6][7][8][9][10][11]. Erhan et al [12] found T lymphocyte domination in the tissue of IGM patients.…”

Section: Introductionmentioning

confidence: 99%

The alteration of lymphocyte subsets in idiopathic granulomatous mastitis

Emsen¹,

Köksal²,

Uçaryılmaz³

et al. 2021

Turk J Med Sci

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Background and aim: This study analyzed peripheral blood lymphocyte subsets to determine their role in the etiopathogenesis of IGM. Materials and methods:This study includes 51 pathologically proven IGM patients (active disease: 26 and in remission: 25) and 28 healthy volunteers. The analyses of lymphocyte subsets were performed by flow cytometric immunophenotyping. Results:The percentage of T helper lymphocyte of all IGM patients were lower than control groups (p = .001). Absolute cytotoxic T lymphocyte count (p=.03), both percentage (p=.035) and absolute count (p=.002) of the natural killer cells, and both percentage (p=.038) and absolute count (p=.008) of natural killer T cells, were higher than the control group. The T helper lymphocyte percentage of the patients with active disease was lower than the control group (p = .0003). The absolute cytotoxic T lymphocyte (p=.029) and natural killer T cells (p=.012) of the patients with active disease were higher than the control group. Conclusion:Idiopathic granulomatous mastitis is defined as a localized form of granulomatous disorders. However, the observed changes in T cells, NK and NKT cells suggest that there is systemic immune dysregulation in patients with IGM.

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Idiopathic granulomatous mastitis with erythema nodosum and polyarthritis

Cited by 20 publications

References 7 publications

Coexistence of idiopathic granulomatous mastitis and erythemanodosum: successful treatment with corticosteroids

Coexistence of idiopathic granulomatous mastitis and erythemanodosum: successful treatment with corticosteroids

Idiopathic granulomatous mastitis with erythema nodosum: Is it a variant of clinical presentation indicating treatment resistance? A retrospective cohort study

The alteration of lymphocyte subsets in idiopathic granulomatous mastitis

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