[A case of chronic rhinosinusitis with nasal polyp with comorbid respiratory epithelial adenomatoid hamartoma with poor response to biologics].
Researchers
Jingyu Jiang, Shen Shen, Mu Xian, Chengshuo Wang, Luo Zhang
Abstract
<b>Objective:</b>This paper reports a case of a male patient who complained about nasal congestion and loss of smell for 20 years. Physical examination revealed polyp-like masses in bilateral olfactory clefts and middle meatus. The patient was diagnosed with chronic rhinosinusitis with nasal polyps(CRSwNP) and exhibited poor response to an adequate course of topical intranasal corticosteroids, then he enrolled in two clinical trials using anti IL4-Rα and anti-TSLP monoclonal antibodies consequently. The treatment duration for each trial was 16 weeks, respectively. The size of his nasal polyps did not change, which was the main endpoint result for both clinical trials. Then the patient underwent an endoscopic surgery and the pathology was respiratory epithelial adenomatoid hamartoma(REAH), which was further confirmed by the single cell sequencing results of the operative specimen, which were different from normal nasal polyps. The diagnosis of REAH is dependent on pathology and preoperative examination has limited specificity, and REAH may share similarities with nasal polyps. Therefore, REAH should be considered for the differential diagnosis when patients with polyp-like masses exhibit poor response to sufficient treatment using biologics. <b>目的:</b>本文报道1例男性患者,因“鼻塞伴嗅觉减退20余年”就诊,体检可见双侧中鼻道及嗅裂区息肉样肿物,足量鼻喷激素治疗效果不佳,先后予白细胞介素4受体α亚基(IL4-Rα)重组人源化单克隆抗体治疗16周及抗胸腺基质淋巴细胞生成素(TSLP)单克隆抗体治疗16周,治疗后鼻息肉大小无明显改变,行鼻内镜手术治疗,术后病理提示鼻腔呼吸上皮腺瘤样错构瘤。因鼻腔呼吸道上皮腺瘤样错构瘤临床检验检查无特异性,可与普通鼻息肉表现类似,确诊需依赖病理,提示在生物制剂治疗慢性鼻窦炎伴鼻息肉患者疗效不佳时,临床医生需提高对于患者合并本病的警惕。.Source: PubMed (PMID: 42402688)View Original on PubMed