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摘要: 目的 分析鼻腔呼吸上皮腺瘤样错构瘤(respiratory epithelial adenomatoid hamartoma,REAH)病理及临床特点,总结诊断要点,提高该病诊治经验。方法 回顾性分析16例REAH的临床资料,总结该病的临床表现、病理学特点、影像学特征、手术治疗方式及预后。结果 16例鼻腔REAH患者,同时伴有鼻窦炎10例(62.50%),伴有内翻性乳头状瘤1例(6.25%),伴有血管瘤1例(6.25%);5例(31.25%)患者有鼻腔鼻窦手术史,其中1例有3次鼻腔鼻窦手术史,1例有2次鼻腔鼻窦手术史,3例有1次鼻腔鼻窦手术史;发生于双侧嗅裂10例(62.50%),单侧嗅裂2例(12.50%),单侧中鼻甲3例(18.75%),鼻咽部1例(6.25%)。16例患者均经病理诊断为REAH,其中病变位于双侧嗅裂患者的术前鼻窦CT上均可观察到嗅裂对称性增宽及中鼻甲外移,双侧嗅裂宽度平均为(9.90±2.70) mm,宽窄嗅裂比值为1.21±0.19,两侧鼻窦Lund-Mackay评分差异无统计学意义。患者手术均在全身麻醉鼻内镜下完成,随访1~66个月,均无复发。结论 结合临床表现及内镜检查和影像学特征有助于REAH的术前诊断,通过内镜下的完整切除可以达到良好的治疗效果。Abstract: Objectives To analyze the pathological and clinical features of nasal respiratory epithelial adenomatoid hamartoma(REAH), and summarize the diagnostic points, to improve the experience of diagnosis and treatment.Methods The clinical data of 16 patients with REAH were analyzed retrospectively. The clinical manifestations, pathological features, imaging features, surgical treatment and prognosis were summarized.Results 16 cases of REAH were studied, 10 cases(62.50%) were associated with sinusitis, 1 case(6.25%) was associated with inverted papilloma, 1 case(6.25%) was associated with hemangioma. 5 cases(31.25%) had a history of nasal sinus surgery, including 1 case with 3 times of nasal sinus surgery, 1 case with 2 times of nasal sinus surgery, 3 cases with 1 time of nasal sinus surgery; 10 cases(62.50%) occurred in the bilateral olfactory cleft, 2 cases(12.50%) in the unilateral olfactory cleft, 3 cases(18.75%) in the unilateral middle turbinate, 1 case(6.25%) in the nasopharynx. All 16 patients were pathologically diagnosed as REAH. In the patients with lesions located in bilateral olfactory fissures, symmetrical widening of olfactory fissures and lateral displacement of middle turbinate were observed on preoperative sinus CT. The average width of bilateral olfactory fissures was (9.9±2.70) mm. The ratio of wide to narrow olfactory cleft was 1.21 ± 0.19. There was no significant difference in Lund-Mackay score between the two sides(P>0.05). All patients underwent surgery under general anesthesia and nasal endoscopy. The follow-up period ranged from 1 to 66 months, and no recurrence occurred.Conclusion Preoperative diagnosis of REAH is facilitated by the combination of clinical manifestations and endoscopic and imaging features. Endoscopic complete resection can achieve a good therapeutic effect.
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Key words:
- nasal cavity /
- hamartoma /
- diagnosis /
- endoscopic surgery
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[1] Nguyen DT, Nguyen-Thi PL, Gauchotte G, et al. Predictors of respiratory epithelial adenomatoid hamartomas of the olfactory clefts in patients with nasal polyposis[J]. Laryngoscope, 2014, 124(11): 2461-2465. doi: 10.1002/lary.24778
[2] Hawley KA, Pabon S, Hoschar AP, et al. The presentation and clinical significance of sinonasal respiratory epithelial adenomatoid hamartoma(REAH)[J]. Int Forum Allergy Rhinol, 2013, 3(3): 248-253. doi: 10.1002/alr.21083
[3] Lima NB, Jankowski R, Georgel T, et al. Respiratory adenomatoid hamartoma must be suspected on CT-scan enlargement of the olfactory clefts[J]. Rhinology, 2006, 44(4): 264-269.
[4] Ozolek JA, Hunt JL. Tumor suppressor gene alterations in respiratory epithelial adenomatoid hamartoma(REAH): comparison to sinonasal adenocarcinoma and inflamed sinonasal mucosa[J]. Am J Surg Pathol, 2006, 30(12): 1576-1580. doi: 10.1097/01.pas.0000213344.55605.77
[5] Kumar D, Handa KK, Handa A, et al. Recurrent respiratory epithelial adenomatoid hamartoma of the nasal cavity[J]. Proc(Bayl Univ Med Cent), 2022, 35(5): 668-669.
[6] Mladina R, Skitarelic N, Poje G, et al. Bilateral respiratory epithelial adenomatoid hamartoma of the olfactory cleft penetrating into the endocranium[J]. J Craniofac Surg, 2011, 22(5): 1905-1907. doi: 10.1097/SCS.0b013e31822ea68b
[7] Nguyen DT, Nguyen-Thi PL, Gauchotte G, et al. Predictors of respiratory epithelial adenomatoid hamartomas of the olfactory clefts in patients with nasal polyposis[J]. Laryngoscope, 2014, 124(11): 2461-2465. doi: 10.1002/lary.24778
[8] Nguyen DT, Jankowski R, Bey A, et al. Respiratory Epithelial Adenomatoid Hamartoma is Frequent in Olfactory Cleft After Nasalization[J]. Laryngoscope, 2020, 130(9): 2098-2104. doi: 10.1002/lary.28298
[9] Nguyen DT, Gauchotte G, Arous F, et al. Respiratory epithelial adenomatoid hamartoma of the nose: an updated review[J]. Am J Rhinol Allergy, 2014, 28(5): 187-192. doi: 10.2500/ajra.2014.28.4085
[10] Seol JG, Livolsi VA, O'Malley BW Jr, et al. Respiratory epithelial adenomatoid hamartoma of the bilateral olfactory recesses: a neoplastic mimic?[J]. AJNR Am J Neuroradiol, 2010, 31(2): 277-279. doi: 10.3174/ajnr.A1755
[11] Li R, Saluja K, Lin M, et al. Sinonasal Hamartomas: From Nasal Chondromesenchymal Hamartoma to Respiratory Epithelial Adenomatoid Hamartoma. Report of six Cases and Review of the Literature[J]. Int J Surg Pathol, 2022, 30(4): 448-456. doi: 10.1177/10668969211064211
[12] 许庆庆, 张媛, 段甦, 等. 鼻腔呼吸上皮腺瘤样错构瘤临床和病理特征分析[J]. 中国耳鼻咽喉头颈外科, 2022, 29(8): 507-510. https://www.cnki.com.cn/Article/CJFDTOTAL-EBYT202208007.htm
[13] Safi C, Li C, Tabaee A, et al. Outcomes and imaging findings of respiratory epithelial adenomatoid hamartoma: a systematic review[J]. Int Forum Allergy Rhinol, 2019, 9(6): 674-680. doi: 10.1002/alr.22298
[14] 付琳, 刘鹏涛, 杨本涛, 等. 鼻腔嗅裂区呼吸上皮腺瘤样错构瘤的CT和MRI表现[J]. 中华放射学杂志, 2016, 50(4): 256-259.
[15] de Saint Hilaire T, Rumeau C, Gallet P, et al. Difference between respiratory epithelial adenomatoid hamartomas and small malignant tumours of the olfactory cleft on CT scans in forty-six patients[J]. Clin Otolaryngol, 2017, 42(6): 1421-1425. doi: 10.1111/coa.12930
[16] Hawley KA, Ahmed M, Sindwani R. CT findings of sinonasal respiratory epithelial adenomatoid hamartoma: a closer look at the olfactory clefts[J]. AJNR Am J Neuroradiol, 2013, 34(5): 1086-1090. doi: 10.3174/ajnr.A3345
[17] 于晓峰, 赵鹤, 曹志伟. 嗅裂区呼吸道上皮腺瘤样错构瘤的影像分析[J]. 临床耳鼻咽喉头颈外科杂志, 2018, 32(12): 924-926. https://www.cnki.com.cn/Article/CJFDTOTAL-LCEH201812011.htm
[18] 陈晴, 戴嵩. 鼻腔嗅裂区呼吸道上皮腺瘤样错构瘤的影像学分析[J]. 临床耳鼻咽喉头颈外科杂志, 2019, 33(6): 557-560. https://www.cnki.com.cn/Article/CJFDTOTAL-LCEH201906022.htm
[19] Braun JJ, Riehm S, Averous G, et al. MRI in respiratory epithelial adenomatoid hamartoma of nasal cavities[J]. J Neuroradiol, 2013, 40(3): 216-219. doi: 10.1016/j.neurad.2012.04.002
[20] Lorentz C, Marie B, Vignaud JM, et al. Respiratory epithelial adenomatoid hamartomas of the olfactory clefts[J]. Eur Arch Otorhinolaryngol, 2012, 269(3): 847-852. doi: 10.1007/s00405-011-1713-9
[21] Boulanger N, Grosjean R, Jankowski R. Pathology of tumours originating in the olfactory cleft[J]. B-ENT, 2011, 17: 21-25.
[22] Al Hawat A, Mouchon E, De Bonnecaze G, et al. Our experience with respiratory epithelial adenomatoid hamartomas of the olfactory cleft[J]. Eur Arch Otorhinolaryngol, 2015, 272(10): 2867-2870.