Abstract
Bronchial fibroepithelial polyps are exceedingly rare with few cases have been reported. They can manifest with a wide array of symptoms; ranging from being totally asymptomatic, cough, refractory dyspnea, and hemoptysis. In our case, our patient’s condition was diagnosed and was managed as asthma. It is one of the rare benign conditions to be encountered, shares similar morphology with other tumors such as angiomyofibroblastoma, aggressive angiomyxoma, and cellular angiofibroma. These lesions have a slow growth pattern which may end up with obstruction. According to the tumor size and symptoms caused by it, treatment varies from observation to complete resection. This case describes an incidental finding of fibroepithelial polyp in the main bronchus for a patient with long-term refractory cough for 5 years, was misdiagnosed to have asthma. Diagnosis typically involves imaging and bronchoscopy, followed by appropriate therapeutic measures and careful monitoring to assess the prognosis.
Introduction
Benign bronchial tracheal lesions are quite uncommon, make up less than 2% of all airway lesions, and have an estimated prevalence in the United State of America of 2.6 per 100 000 habitants. One of those benign tumors is fibroepithelial polyp (FEP), which is hardly found as an endobronchial tumor. 1 In fact, FEPs are commonly found either in the skin or genitourinary tract. Most of these tumors are seen in men between 40 and 70 years of age. 2
These lesions with slow growth rate usually are correlated to a bronchial obstruction and may be present with airway stenosis which end up with bronchiectasis and/or atelectasis. Frequently presented manifestations are refractory asthma, recurrent infection, hemoptysis, and dyspnea. Also, these symptoms are the main cause of hospitalization. 3
In addition, they mirror morphological characteristics akin to those seen in other mesenchymal tumors such as cellular angiofibroma, aggressive angiomyxoma, and angiomyofibroblastoma. 4 There are many different histological manifestations, but the most prevalent one is formed up of collagen fibers in a stroma that is covered by healthy respiratory epithelium. 5 It was described as having solid, spherical, whitish polypoid lesions with a smooth, glistening surface during bronchoscopy for the purpose of diagnosis. 6 Treatment options are contingent upon the size and symptomatology of the lesions. In instances of small lesions, which are often devoid of symptoms, management typically involves corticosteroids and antibiotics, while bigger lesions are typically treated with invasive methods such lobectomy or bronchoscopic polyp removal. 3
However, the pathogenesis of FEP remains poorly understood, which can make it a challenge for accurate diagnosis. According to certain studies, persistent inflammatory processes may be a crucial factors, such as asthma, chronic obstructive pulmonary disease, smoking, infections, foreign body aspiration, and prolonged mechanical ventilation. 7
Herein, we present a rare case of bronchial FEP presented with long-term cough and misdiagnosed as asthma, with review of the literature.
Case Presentation
Our patient is a 38-year-old male from Nablus, Palestine, who sought our medical advice because of persistent productive cough for 5 years. He had occasional hemoptysis. No history of fever, shortness of breath, heartburn, dysphagia, or weight loss, he denies any episode of aspiration. He works as a barber and has insignificant past medical and surgical history. His family history was unremarkable. He is a smoker with 38 pack-year smoking history. The patient sought the first medical advice on 2018, then he was followed up by several physicians, he was diagnosed to have asthma and was given inhalers and antihistamines as treatment, but there was no improvement in spite of being adherent to the treatment. On examination, the patient was doing well, not in respiratory destress, chest auscultation revealed a reduced air entry with expiratory wheezes of the right lung, but normal air entry with no added sounds of the left, his blood oxygen saturation was 97% in room air. Otherwise, physical examination was unremarkable.
Multiple investigations were done, including complete blood count (CBC), coagulation study, and blood chemistry tests, which all showed normal results. According to his history and clinical findings, we decided to do chest computed tomography (CT) scan, which revealed a right endobronchial lesion that had partially obstructed the bronchus intermedius, which resulted in partial collapse of the right middle and lower lobes (Figure 1).
Chest computed tomography (CT) scan shows a multiple intra bronchial soft tissues (arrow) in the right lung, one of them seen narrowing the upper lobe bronchus, other tissue critically narrowing the bronchus intermedius, resulting in partial collapse of the right middle and lower lobes: (A) coronal view and (B) axial view.
After that, the patient underwent flexible bronchoscopy with multiple hot snaring of a huge sessile endobronchial lesion located in bronchus intermedius, causing a subtotal obstruction, the lesion was totally removed (Figures 2 and 3). Biopsy was taken, histopathological examination concluded a lesion with features of FEP, no evidence of malignancy was seen (Figure 4). Four months later, a follow-up flexible bronchoscopy was done and revealed a normal bronchial anatomy, with no evidence of residual tissue or local recurrence.
Bronchoscopy view showing a huge sessile lesion obstructing the right bronchus.
FEP pieces after excision.
Histopathological evaluation: A polyp covered by benign pseudostratified ciliated columnar (respiratory epithelium). (A) There is a polypoid mass with midly fibrocellular stroma and a sparse lymphocytic infiltrate. (B) Prominent dilated small thin walled vessels associated with fibro collagenous stroma and mildly acanthotic mucosa. No dysplasia or malignancy was notice.
Discussion
Bronchial FEPs are rarely encountered in clinical practice, initially described by Rowlands 8 in 1960. In the English literature, only a few cases are reported. 9 After reviewing existing studies, we have found that we still do not have a clear understanding of how problematic these polyps can be. Over the last few decades, around 45 cases of tracheobronchial FEP have been recorded. However, we could only access 37 case reports, including our own (Table 1).3,5,7,9-16 Based on what these case reports have suggested, it is been theorized that the development of bronchial FEP might be linked to ongoing irritation of the bronchial passages due to different factors like smoking and infections.
Summary of Prior Case Reports, Including Ours, by Comparing 6 Key Parameters: Age, Pender, Presenting Symptoms, Lesion Size (Measured in Millimeters), Anatomical Location, and the Therapeutic Approach.
Abbreviations: M, male; F, female; ND, no data; RB, right bronchus; LB, left bronchus.
The cause behind bronchial FEP remains unclear. In broad terms, benign bronchial polyps can be categorized as either inflammatory or noninflammatory. However, the precise classification of FEP remains debatable. Certain studies categorize them as inflammatory in nature, while an alternate perspective proposes that the inflammation arises due to sporadic obstruction within the respiratory tract induced by the polyp’s mass effect. 10
The deferential diagnostic possibilities for FEP lesions encompass neoplastic conditions (metastatic lesions in the airway, bronchogenic carcinoma, plasmacytomas), infectious origins (papilloma virus, mucus plug), rheumatological disorders (vasculitis, rheumatoid arthritis, amyloidosis), as well as benign airway anomalies (central airway granulomas, chondromas, neuromas, leiomyomas, lipomas, hamartomas). Histopathological scrutiny unveiled the existence of a polyp characterized by a covering of regular respiratory epithelium, wherein a confined presence of long-term inflammatory infiltration was discernible, primarily composed of lymphocytes and plasma cells. A range of long-term inflammatory origins, such as foreign body aspiration, extended mechanical ventilation, asthma, persistent exposure to smoke, long-term sinusitis, and mycobacterial infections, potentially represent underlying factors in the genesis of these findings. 9
A case report was published in 66-year-old female came with a primary complaint of hemoptysis persisting for 2 days. A CT scan revealed the presence of bronchiectasis, ground glass opacities in the lower lobes, and thickening of left main bronchus wall. Subsequent biopsy specimens confirmed the presence of FEP. 3
A study was done in 2017 collected from 15 cases, the symptoms vary between asymptomatic, cough, fever, hemoptysis and pneumonia-like opacity, some cases presented solely with a recurrent dry cough. Irrespective of its size, all cases exhibited consistent histological features during bronchoscopy. The lesion consistently manifested as a solid endobronchial nodule with a firm, whitish, smooth, and glistening surface. In some instances, particularly with the largest polyps, a multilobulated and septimated surface was observed, Notably. 6
Another case report of a 48-year-old woman presented with persistent cough and fever, polyp-like lesion found on CT scan and bronchoscopy that has obstructed the left main bronchus. Surgical excision with biopsy was performed, the biopsy showed FEP. 17
A case report published in 2017 for 79-year-old smoker male with 3 months history of dry cough as the main complaint, on CT scan a 6-mm polyp found in the proximal right side of the trachea. Bronchoscopy with biopsy was done and showed a polyp with a marked papillary projections and long stalk, revealing an FEP. 18
A separate case study conducted in the year 2020 centered around a 61-year-old male who had a history of smoking. Upon hospital admission, he presented with a moderate fever alongside a nonproductive cough that had persisted for a week. Following the CT imaging, it became evident that there was consolidation and atelectasis observed within the right lower lobe. In addition, there was an obstruction observed at the anatomical level of the right pulmonary artery, specifically affecting the right intermediate bronchus. Subsequent bronchoscopy revealed the presence of a pedunculated and pinkish polypoid tumor within the bronchus of the right lower lobe, notable for its protruding. Remarkably, no indications of bronchial wall invasion were detected during this procedure. Detailed pathological examination of the specimen ultimately confirmed the presence of FEP. 7
The lesions ranged from 2 to 100 mm of maximum diameter. There are eighteen lesions measuring 10 mm or more. Symptoms were generally mild or occurred in conjunction with pneumonia. Among the evaluated cases, 21.5% (8 out of 37) exhibited hemoptysis, 1 case encountered airway obstruction, and 2 cases were characterized by productive coughing. A number of cases were identified incidentally through radiological scans associated with pneumonia (11 cases), or were discovered in 8 patients who displayed no symptoms. According to our case, the initial presentation was long-term cough for 5 years, other cases were presented with different symptoms, long-term productive cough not being mentioned as the main complaint.
In addition, the average age at the time of diagnosis was 60 years, with a standard deviation of ±14.34 years. The occurrence of this condition was less frequent among women, accounting for 19% of the total 37 cases.
Radiologic results generally demonstrate a clearly defined polyp growth is visible in the chest CT scan, and the edges of the polyp have a lobulated appearance. 1 The dimensions and locations of focal pulmonary FEP exhibit notable diversity. Our observations indicated a mean diameter of 31 mm (with a standard deviation of 18.1 mm). Among the total of 37 cases, these lesions were distributed as follows: 48.6% (18 cases) were situated in the right bronchial tree, 37.8% (14 cases) in the left bronchial tree, and 13.6% (5 cases) in the trachea. A comprehensive assessment of the clinical, radiological, and therapeutic strategies employed in the documented cases has been succinctly consolidated and presented in the provided Table 1.
In the case presented, what initially appeared as a straightforward asthma turned into a complex diagnostic challenge with profound implications. The patient’s clinical history included symptoms suggestive of asthma, leading to a series of misdiagnoses over a span of 4 years till he recently suffered from mild hemoptysis. Therefore, we did further investigations, that have revealed an airway lesion. This unexpected twist underscores the importance of considering a wide range of differential diagnosis.
Finally, there is a lack of documented mortality data attributed to FEP, and no instances of mortality related to treatment interventions have been recorded. Consequently, the prognosis is largely good. Remarkably, no cases of FEP undergoing malignant transformation have been identified in existing reports. For such cases, the recommended therapeutic approach involves the utilization of endobronchial resection techniques, employing mechanical debulking, laser therapy, or electrocautery. Surgical intervention is infrequently warranted and reserved for circumstances where the feasibility of endobronchial resection is compromised or when pathological interpretations introduce ambiguity. 19 As for our patient, the treatment was bronchoscopy with laser removal of the endobronchial tumor. As far as our current understanding goes, other published reports have not provided any evidence of recurrence, with the exception of a single case published in 2013 by Ming Li and colleagues. 7 Three months subsequent to the initial assessment, a follow-up bronchoscopy was conducted to evaluate the patient’s condition. The ensuing findings provided compelling evidence supporting the absence of tumor recurrence.
Conclusion
Fibroepithelial polyps of the bronchus are rare and have variable presenting symptoms. As far as we can tell, 45 case reports were published throughout the previous decades, not all are available for analysis. Bronchial FEPs share a similar morphological characteristic with other pathologies of the airways. In addition to the lack of well-defined and reliable diagnostic criteria based on its appearance, misdiagnosis may occur, leading to potential confusion with other bronchial conditions. The treatment is mainly by endobronchial resection of the lesion, with negligible risk for recurrence.
Footnotes
Acknowledgements
The completion of this case could not have been possible without the participation and assistance of many people whose names may not be enumerated like our patient’s parents, whose contribution are sincerely appreciated and gratefully
Declaration of Conflicting Interests
The author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.
Funding
The author(s) received no financial support for the research, authorship, and/or publication of this article.
Ethics Approval
Our institution does not require ethical approval for reporting individual cases or case series.
Informed Consent
Verbal informed consent was obtained from the patient(s) for their anonymized information to be published in this article.
