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肉芽肿性乳腺炎、结节性红斑和多发性关节炎:一例报告。

Granulomatous mastitis, erythema nodosum, and polyarthritis: a case report.

机构信息

Flushing Hospital Medical Center, 4500 Parsons Boulevard, Flushing, NY, 11355, USA.

Pediatric Rheumatology Marshfield Clinic, 1000 North Oak Ave, Suite 1A1,, Marshfield, WI, 5449, USA.

出版信息

J Med Case Rep. 2022 Apr 5;16(1):146. doi: 10.1186/s13256-022-03327-5.

Abstract

BACKGROUND

Granulomatous mastitis is a rare inflammatory disease of the breast, typically seen in woman of child-bearing age. No definitive etiology has been described. In rare instances, this condition has been reported to be associated with extramammary manifestations such as erythema nodosum and arthritis. We describe this rare condition in an adolescent female.

CASE PRESENTATION

A 16-year-old, Hispanic female presented with right-sided painful breast swelling, polyarthritis, and erythema nodosum on bilateral shins and lower thighs. Physical examination was negative for lymphadenopathy and pulmonary, gastrointestinal, and cardiovascular findings. Ophthalmologic examination for uveitis and serologic tests for autoimmune diseases were negative. Diagnosis of idiopathic granulomatous mastitis was made by exclusion of other etiologies and conditions. Confirmation was made by histopathologic examination demonstrating noncaseating granuloma within breast lobules with neutrophils and microabscess formation. After wide local excision and a short course of trimethoprim-sulfamethoxazole, our patient was placed on naproxen and prednisone, the latter being tapered off over 3 months, with steady and complete resolution of all symptoms.

CONCLUSION

This is the first reported case of idiopathic granulomatous mastitis in a pediatric patient who also had extramammary manifestations, including erythema nodosum and polyarthritis. In this case-based review, we summarize the phenotype, risk factors, prognosis, and treatment options of this rare condition, chiefly to make the readers cognizant of such a diagnostic possibility in similar clinical presentation in the future.

摘要

背景

肉芽肿性乳腺炎是一种罕见的乳腺炎症性疾病,多见于育龄期妇女。目前尚未明确其病因。在极少数情况下,这种疾病与结节性红斑和关节炎等乳腺外表现有关。我们在此报告一例青少年女性患者。

病例介绍

一名 16 岁的西班牙裔女性因右侧乳房疼痛性肿胀、多关节炎和双侧小腿及大腿下部结节性红斑就诊。体格检查无淋巴结肿大和肺、胃肠道及心血管异常。眼科检查排除了葡萄膜炎,自身免疫性疾病的血清学检查也为阴性。通过排除其他病因和疾病,诊断为特发性肉芽肿性乳腺炎。组织病理学检查显示乳腺小叶内有非干酪样肉芽肿,伴有中性粒细胞和微脓肿形成,证实了诊断。患者接受了广泛局部切除术和短期甲氧苄啶-磺胺甲噁唑治疗,随后给予萘普生和泼尼松治疗,泼尼松在 3 个月内逐渐减量,所有症状均稳定且完全缓解。

结论

这是首例报告的儿童特发性肉芽肿性乳腺炎病例,同时存在乳腺外表现,包括结节性红斑和多关节炎。在本病例报告中,我们总结了这种罕见疾病的表型、危险因素、预后和治疗选择,主要是为了使读者在未来遇到类似临床表现时能够认识到这种诊断可能性。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8991/8985372/85d980b163d2/13256_2022_3327_Fig1_HTML.jpg

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