Multiple choroidal granulomas in lymphomatoid granulomatosis: a case report.

IF 1.7 4区 医学 Q3 OPHTHALMOLOGY
Ryo Taguchi, Yoshiaki Tanaka, Machiko Shimmura, Hiroko Takano, Akihiro Kakehashi, Toshikatsu Kaburaki
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Abstract

Background: Lymphomatoid granulomatosis (LYG) is a rare B-cell lymphoproliferative disorder predominantly affecting middle-aged men. The disease occurs primarily in the lungs; however, reports of lesions in intraocular regions are scarce. Herein, we report a case of choroidal granulomas in a patient with LYG.

Case presentation: A 55-year-old male patient presented to a respiratory specialist with pneumonia of unknown cause. His serum-soluble IL-2 receptor level was highly elevated, and transbronchial lung biopsies revealed granulomatous lesions. Sarcoidosis was suspected, and an ophthalmological examination was performed. The best-corrected visual acuity was 1.2 in both eyes. Although no cells were present in the anterior chambers or anterior vitreous in either eye, multiple large yellowish-white exudates on the fundus of the right eye and soft exudates around the optic disc in the left eye were observed. Optical coherence tomography revealed choroidal granulomas consistent with the yellowish-white lesions in the right eye. Fluorescein angiography revealed tissue staining in the right eye consistent with granulomatous choroidal lesions. Both eyes showed capillaritis, mainly in the nasal retina. Subsequently, a thoracoscopic left lung biopsy led to a pathological diagnosis of LYG, and chemotherapy was initiated. Thereafter, the yellowish-white exudates gradually scarred, and the choroidal granulomas and soft exudates disappeared; thus, a vitreous biopsy was not performed. After autologous hematopoietic stem cell transplantation, the LYG was in remission. No ocular lesion recurrence was observed in the subsequent 18 months.

Conclusions: LYG may cause multiple large choroidal granulomas.

类淋巴瘤肉芽肿病多发性脉络膜肉芽肿1例。
背景:类淋巴瘤肉芽肿病(LYG)是一种罕见的b淋巴细胞增生性疾病,主要发生在中年男性。这种疾病主要发生在肺部;然而,关于眼内病变的报道很少。在此,我们报告一例LYG患者的脉络膜肉芽肿。病例介绍:一名55岁男性患者,因不明原因肺炎就诊于呼吸道专科。他的血清可溶性IL-2受体水平高,经支气管肺活检显示肉芽肿病变。怀疑结节病,行眼科检查。双眼最佳矫正视力为1.2。虽然两眼前房和前玻璃体均未见细胞,但右眼眼底可见大量黄白色渗出物,左眼视盘周围可见软渗出物。光学相干断层扫描显示脉络膜肉芽肿与右眼黄白色病变一致。荧光素血管造影显示右眼组织染色与肉芽肿性脉络膜病变一致。双眼毛细血管炎,以鼻视网膜为主。随后,胸腔镜左肺活检病理诊断为LYG,并开始化疗。此后黄白色渗出物逐渐结疤,脉络膜肉芽肿及软渗出物消失;因此,没有进行玻璃体活检。自体造血干细胞移植后,LYG得到缓解。术后18个月未见眼部病变复发。结论:LYG可引起多发性大脉络膜肉芽肿。
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来源期刊
BMC Ophthalmology
BMC Ophthalmology OPHTHALMOLOGY-
CiteScore
3.40
自引率
5.00%
发文量
441
审稿时长
6-12 weeks
期刊介绍: BMC Ophthalmology is an open access, peer-reviewed journal that considers articles on all aspects of the prevention, diagnosis and management of eye disorders, as well as related molecular genetics, pathophysiology, and epidemiology.
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