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14岁共济失调毛细血管扩张症男性患者中表现为急性泪囊炎的伯基特淋巴瘤:病例报告及文献复习

Burkitt's lymphoma presenting as acute dacryocystitis in a 14-year-Old male with ataxia telangiectasia: A case report and review of the literature.

作者信息

Khannejad Samin, Mansourian Sajad, Aghajani Amirhossein, Nozarian Zohreh, Rafizadeh Seyed Mohsen

机构信息

School of Medicine, Shahid Beheshti University of Medical Sciences, Tehran, Iran.

Department of Oculo-facial Plastic and Reconstructive Surgery, Farabi Eye Hospital, Tehran University of Medical Sciences, Tehran, Iran.

出版信息

Am J Ophthalmol Case Rep. 2025 Feb 24;38:102288. doi: 10.1016/j.ajoc.2025.102288. eCollection 2025 Jun.

Abstract

PURPOSE

This report describes an atypical presentation of a case of Burkitt lymphoma in a pediatric patient with ataxia telangiectasia.

MAIN OBSERVATIONS

A 14-year-old boy with a history of AT was referred to our hospital with lower eyelid swelling and medial canthus abscess. On physical examination, movement of the left eye was limited in downgaze and adduction. Two mm proptosis was observed on the left side. Snellen's Visual acuity in the left eye was 8/20. The rest of the examination was normal. Magnetic resonance imaging (MRI) was performed and showed an infiltrative mass in the inferior of the left orbit, left ethmoidal sinus, and maxillary sinus. The patient subsequently underwent an incisional biopsy. The biopsy specimen was sent for histopathologic evaluation. Histopathology was significant for atypical monotonous cell infiltrations in the fibroconnective tissue and the presence of pleomorphic, irregularly shaped nuclei with multiple mitoses. Immunohistochemistry (IHC) findings were consistent with Burkitt's lymphoma, and the patient was referred to the oncology department for chemotherapy and appropriate treatment.

CONCLUSION

and SignificanceOrbital and lacrimal duct involvement is a rare presentation of Burkitt lymphoma especially in the pediatric population. In the new-onset nasolacrimal duct obstruction (NLDO) in a child or teenager, the differential diagnosis should include malignancies, especially leukemia/lymphoma infiltrations.

摘要

目的

本报告描述了一名患有共济失调毛细血管扩张症的儿科患者的伯基特淋巴瘤的非典型表现。

主要观察结果

一名有共济失调毛细血管扩张症病史的14岁男孩因下眼睑肿胀和内眦脓肿被转诊至我院。体格检查发现,左眼向下注视和内收时运动受限。左侧观察到2毫米的眼球突出。左眼的斯内伦视力为8/20。其余检查均正常。进行了磁共振成像(MRI)检查,结果显示左侧眼眶下部、左侧筛窦和上颌窦有浸润性肿块。患者随后接受了切开活检。活检标本送去进行组织病理学评估。组织病理学显示纤维结缔组织中有非典型的单调细胞浸润,存在多形性、形状不规则的核以及多个有丝分裂象。免疫组织化学(IHC)结果与伯基特淋巴瘤一致,该患者被转诊至肿瘤科进行化疗及适当治疗。

结论

眼眶和泪道受累是伯基特淋巴瘤的一种罕见表现,尤其是在儿科人群中。对于儿童或青少年新发的鼻泪管阻塞(NLDO),鉴别诊断应包括恶性肿瘤,尤其是白血病/淋巴瘤浸润。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/c09d/11923752/2ff6c22c3e98/gr1.jpg

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