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1例表现为广泛蝇蛆病的结外自然杀伤/T细胞淋巴瘤病例报告

A case report of Extranodal natural killer / T-cell lymphoma presenting as extensive myiasis.

作者信息

Chih Wong Eugene Hung, Peng Quah Gabriel Xia, Yong Doh Jeing

机构信息

Department of Otorhinolaryngology, Faculty of Medicine and Health Sciences, Universiti Malaysia Sabah, Kota Kinabalu, Sabah, Malaysia.

Department of Otorhinolaryngology - Head and Neck Surgery, Queen Elizabeth Hospital, Kota Kinabalu, Sabah, Malaysia.

出版信息

Ann Med Surg (Lond). 2022 Mar 2;75:103419. doi: 10.1016/j.amsu.2022.103419. eCollection 2022 Mar.

Abstract

We described a 56-year-old Indonesian man with one-month history of nasal obstruction and rapidly increasing nasal swelling and two weeks history of severe facial pain with foul smelling discharge, bleeding and noticing live maggots emerging from his nasal and oral cavity. On examination, he appeared cachexic with a markedly swollen, erythematous and deformed external nose. Live maggots, pus and necrotic tissues were found in both nasal cavities, with erosion of upper gingiva and hard palate. Patient was managed initially with tracheostomy under local anaesthesia, followed by removal of around 350 live maggots from the nasal cavities and debridement of necrotic tissues. A midline nasal cavity mass which extended laterally into the nasal cavity was found, along with a large defect over the gingival labial sulcus with necrotic hard palate and a communication between oral and nasal cavities. Biopsies were taken from the nasal mass, gingiva and hard palate. Histopathological results from the biopsies showed diffuse, aggressive infiltrative malignant lymphoid cells with widespread angionecrosis, consistent with features of extranodal NK/T-cell lymphoma. To our knowledge, there is only one other reported case where the diagnosis of ENKTCL was made after patient presented with oro-nasal myiasis. Clinicians should have raised awareness on this atypical presentation so that further investigation and management can be implemented promptly.

摘要

我们描述了一名56岁的印度尼西亚男子,有1个月的鼻塞病史,鼻腔肿胀迅速加重,还有2周的严重面部疼痛病史,伴有恶臭分泌物、出血,且发现有活蛆从他的鼻腔和口腔中爬出。检查时,他看起来消瘦,外鼻明显肿胀、发红且变形。两个鼻腔均发现有活蛆、脓液和坏死组织,上牙龈和硬腭有糜烂。患者最初在局部麻醉下进行了气管切开术,随后从鼻腔中取出约350条活蛆,并对坏死组织进行清创。发现鼻腔中线有一肿物,向外侧延伸至鼻腔,同时牙龈唇沟处有一大的缺损,伴有硬腭坏死,口腔与鼻腔相通。对鼻腔肿物、牙龈和硬腭进行了活检。活检的组织病理学结果显示弥漫性、侵袭性浸润的恶性淋巴细胞,伴有广泛的血管坏死,符合结外NK/T细胞淋巴瘤的特征。据我们所知,仅有另一例报告病例,患者在出现口鼻蝇蛆病后被诊断为结外NK/T细胞淋巴瘤(ENKTCL)。临床医生应提高对这种非典型表现的认识,以便能够及时进行进一步的检查和治疗。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/e6f2/8977926/9bc048b51822/gr1a.jpg

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