气管支气管支气管病骨软骨增生:一例罕见的非吸烟者和非特应性患者,从小就有很长的喘息史。

IF 2.3
Multidisciplinary Respiratory Medicine Pub Date : 2016-04-19 eCollection Date: 2016-01-01 DOI:10.1186/s40248-016-0050-7
Alessandro G Fois, Antonella Arcadu, Luigi Santoru, Rocco Trisolini, Vincenzo Marras, Giorgio C Ginesu, Sara Canu, Lorenzo Cordero, Gabriella Diana, Pietro Pirina
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引用次数: 18

摘要

背景:气管支气管病骨软骨成形性(TBPO)是一种罕见的良性疾病,其特征是气管支气管树粘膜下层出现1-3毫米骨性或化生的软骨结节。气管后膜壁通常不受影响。软骨硬化和外生性结节可引起慢性炎症和粘膜化生、僵硬和气道阻塞。这种疾病通常无症状或伴有非特异性症状,其患病率被低估,诊断时的平均年龄为50岁。病例介绍:我们报告一例49岁男性,非吸烟者。他是一名铁匠,无家可归,出生在罗马尼亚,自幼被诊断患有哮喘。患者因低烧、多汗、咳嗽、脓性痰、高分辨率计算机断层扫描(HRCT)发现主支气管不规则的毛玻璃样混浊而入院。纤维支气管镜检查显示在段性支气管入口处存在粘膜不规则。组织学检查显示骨软骨性结节,与TBPO一致。支气管肺泡灌洗液的微生物学检查也显示铜绿假单胞菌感染。结论:TBPO是一种罕见的疾病,以喘息、咳嗽、咯血和反复肺部感染为特征,典型发病于成年期。在报告的病例中,这些症状始于童年,尽管它们被误解为哮喘。即使在文献中没有关于儿童期发病的报道,也很可能在生命的最初几年发生微小的变化,并在成年后变得更加明显。受累的节段支气管和亚节段支气管,通常在TBPO中,可以解释存在喘息和非生产性咳嗽报告自童年以来的病人。
本文章由计算机程序翻译,如有差异,请以英文原文为准。

Tracheobronchopathia Osteochondroplastica: a rare case report of a non-smoker and non-atopic patient, with a long history of wheezing since childhood.

Tracheobronchopathia Osteochondroplastica: a rare case report of a non-smoker and non-atopic patient, with a long history of wheezing since childhood.

Tracheobronchopathia Osteochondroplastica: a rare case report of a non-smoker and non-atopic patient, with a long history of wheezing since childhood.

Tracheobronchopathia Osteochondroplastica: a rare case report of a non-smoker and non-atopic patient, with a long history of wheezing since childhood.

Background: Tracheobronchopathia Osteochondroplastica (TBPO) is an uncommon and benign condition characterized by osseous or metaplastic cartilaginous 1-3 mm nodules in the submucosa of the tracheo-bronchial tree. Posterior membranous wall of trachea is typically spared. Ecchondrosis and exostosis nodules can cause chronic inflammation and mucosal metaplasia, stiffness and airway obstruction. The prevalence of this disease, often asymptomatic or associated with nonspecific symptoms, is underestimated, and the mean age at diagnosis is 50 years.

Case presentation: We report a case of a 49 year old male, non-smoker. He was a smith, homeless, born in Romania and reported a diagnosis of asthma since childhood. He was admitted to our Respiratory Unit presenting low-grade fever with profuse sweating, cough, purulent sputum, and ground-glass opacity with irregularity in main bronchi detected by High-Resolution Computed Tomography (HRCT) scan. Fibrobronchoscopy revealed the presence of mucosal irregularities up to the segmental bronchi entrance. Histological examination showed nodules of osseouscartilaginous nature, consistent with TBPO. Microbiological tests of Bronchoalveolar Lavage fluid also revealed an infection by Pseudomonas Aeruginosa.

Conclusion: TBPO is a rare disease characterized by wheezing, cough, hemoptysis, and recurrent pulmonary infections, with typical onset during adulthood. In the case reported, the symptoms began in childhood, although they had been misinterpreted as asthma. Even if childhood-onset is not reported in literature, it is likely that small changes occur in the first few years of life and become more evident in adulthood. The involvement of segmental and sub-segmental bronchi, usually spared in TBPO, could explain the presence of wheezing and non-productive cough reported by our patient since childhood.

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来源期刊
Multidisciplinary Respiratory Medicine
Multidisciplinary Respiratory Medicine Medicine-Pulmonary and Respiratory Medicine
自引率
0.00%
发文量
23
期刊介绍: Multidisciplinary Respiratory Medicine is the official journal of the Italian Respiratory Society - Società Italiana di Pneumologia (IRS/SIP). The journal publishes on all aspects of respiratory medicine and related fields, with a particular focus on interdisciplinary and translational research. The interdisciplinary nature of the journal provides a unique opportunity for researchers, clinicians and healthcare professionals across specialties to collaborate and exchange information. The journal provides a high visibility platform for the publication and dissemination of top quality original scientific articles, reviews and important position papers documenting clinical and experimental advances.
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