Arif Maan, Muhammad Soban

Khan, Javaid Ahmed

Sohail, Amir Humza

Sulaiman, Asna

Waleed, Muhammad

English

eCommons@AKU

eCommons@AKU Section of Pulmonary & Critical Care Department of Medicine 12-2019 Solitary endobronchial papilloma: A rare entity Muhammad Soban Arif Maan Muhammad Waleed Amir Humza Sohail Asna Sulaiman Javaid Ahmed Khan Follow this and additional works at: https://ecommons.aku.edu/pakistan_fhs_mc_med_pulm_critcare  Part of the Pulmonology Commons S120 Journal of the College of Physicians and Surgeons Pakistan 2019, Vol. 29 (Supplement 2): S120-S122INTRODUCTIONSolitary papillomas of the lower airways, also known assolitary endobronchial papillomas (SEPs), are benigntumours that are rarely encountered in the clinic. Theetiology of these neoplasms is not fully understood,although smoking and human papilloma virus (HPV)infection are hypothesised to have a contributory role.1SEPs have been divided for ease of management intothree histologically distinct sub-categories.1 Treatmentfor SEPs remains a grey area primarily because of thedearth of clinical data available due to their rarity of thispathology. Herein, we present the case of a 73-yearmale who presented to us with cough and hemoptysisand was subsequently found to have a solitarysquamous papilloma located in the right bronchus. Wewill also briefly review briefly the main diagnostic andtreatment modalities that have proven effective for SEPsaccording to the existing literature. CASE REPORTA 73-year male presented to the emergency room withcomplaints of cough and hemoptysis for three days. Thehemoptysis was fresh blood, about a quarter cup,roughly four to five times a day with no clots. The patienthad been a smoker for the past 50 years and hadconsistently smoked 3 to 4 packs of cigarettes per day.He was a tobacco chewer as well. He had been usinghomeopathic drugs for several years, along with severalmultivitamin supplements and antibiotics prescribedby various general practitioners. He was a known caseof chronic obstructive pulmonary disease (COPD).Clinically, he looked well, slightly overweight andafebrile. On examination, the chest expansion wasdecreased on both sides, and the percussion note wasnoticeably hyper-resonant on the right side. Right midzone crackles were present and the anteroposterior (AP)diameter was increased. There was normal vesicularbreathing with minimal wheezes bilaterally. His pulsewas regular. Rest of the general physical examinationwas unremarkable.His routine investigations (including complete bloodcount, urea, creatinine and electrolyte levels) werewithin normal limits. Chest X-ray showed hyperinflatedlungs with a prominent right hilum.The patient was managed as a case of exacerbatedCOPD and admitted for further workup. A bronchoscopywas done. The bronchoscopy revealed normal vocalcords with some overlying blood which was aspirated.The right bronchial tree was normal except at the originof the right upper lobe bronchus, where a polypoidalmass measuring 3 × 5 × 4 cm was visualised (Figure 1).Bronchoscopic biopsy of this mass revealed a benignpapillomatous growth composed of squamous epithelialcells which were thrown into papillary projections (Figure 2).Fibrovascular cores showed mild chronic non-specificinflammatory cells. Malignant cells were not seen.Surgical removal of this papilloma was advised but thepatient declined surgery. He, however, agreed uponregular follow-up visits. He was well at 2 months and at6 months follow-up with no recurrence of hemoptysis.  CASE REPORTSolitary Endobronchial Papilloma: A Rare EntityMuhammad Soban Arif Maan1, Muhammad Waleed1, Amir Humza Sohail1, Asna Sulaiman1 and Javaid Ahmed Khan21Medical College, The Aga Khan University, Karachi, Pakistan2Department of Pulmonology and Critical Care, The Aga Khan University, Karachi, PakistanABSTRACTSolitary papillomas of the lower airways are uncommon clinical entities that present a diagnostic challenge because theyare not usually considered among the differential diagnoses. Reliable clinical data about their presentation and treatmentremain scarce. We describe a case of an elderly male who presented to us with cough and hemoptysis. He had been asmoker for 50 years. Chest radiograph revealed hyperinflated lungs and slight hilar enlargement on the right side.Bronchoscopy revealed a polypoid mass at the origin of the right upper lobe bronchus. Biopsy showed it to be apapillomatous growth composed of squamous epithelial cells. No malignant cells were detected microscopically. Patientdeclined surgical removal of this papilloma. He was fine on followup visits during the subsequent months. In patients withhemoptysis, solitary endobronchial papilloma should also be considered in the differential diagnosisKey Words: Papilloma, Endobronchial, Hemoptysis.How to cite this article: Maan MSA, Waleed M, Sohail AH, Sulaiman A, Khan JA. Solitary endobronchial papilloma: A rare entity. J CollPhysicians Surg Pak 2019; 29 (Supplement 2):S120-S122.Correspondence to: Dr. Muhammad Soban Arif Maan,Medical College, The Aga Khan University, Stadium Road,Karachi, PakistanE-mail: sobanmaan@live.co.ukReceived: December 14, 2018;   Revised: April 08, 2019;Accepted: June 13, 2019DISCUSSIONIn developing areas of the world, such as the Indiansubcontinent, tuberculosis is by far the most commoncause of hemoptysis.2 Other frequent etiologies ofhemoptysis are pneumonia, vasculitis, fungal infections,lung cancer, bronchitis, bronchiectasis and congenitalheart disease.2 Because of their rarity, SEPs aregenerally not considered among the differentials. Thismakes their diagnosis challenging.SEPs are very rare; only about a 100 have been reportedin the medical literature.3 They occur with a higherfrequency in men than women (3:1) and the vastmajority are found in tobacco users.1 These tumourshave been allocated three separate categories based onmicroscopic features: Squamous papilloma, glandularpapilloma and mixed papilloma which displays a mixtureof the other two types.1 Out of these, squamouspapillomas have strongest association with tobaccosmoking, and they are also most susceptible tomalignant transformation.4 The patient had a solitarysquamous papilloma of the bronchus. These papillomas arise from the respiratory mucosalepithelium and measure between 0.7 and 2.5 cm indiameter. Grossly, they are soft polypoid lesions that aremostly exophytic, projecting into the airway lumen andnarrowing it. This elevates the risk of post-obstructiveinfections and other complications such as atelectasisand bronchiectasis.5 SEPs may be completely asymp-tomatic incidental findings.6 When symptoms arepresent, they may be cough, wheezing and asthma-likesymptoms, hemoptysis and dyspnea.6 Obstructivesymptoms may also be present. Many SEPs do notshow up on chest X-ray, which can lead to a delay inthe diagnosis.5 CT is the preferred imaging modality andit usually shows a polypoidal nodule that may beobstructing the airway.5 The scan might show obs-tructive pneumonitis and atelectasis when there iscomplete airway obstruction. Partial but significantobstruction leads to reflex vasoconstriction resulting indiminished perfusion; this leads to hyperlucency of theaffected areas.5 Distal airway and parenchymal involve-ment on CT manifests as numerous cavitate nodularlesions.5 In our patient, the papilloma was discoveredduring bronchoscopy which was undertaken to identifythe cause of his hemoptysis. The endoscopic findingsdepend upon the location and size of the lesion.Stenosis might be seen because of the SEP, but therehas been a report of a papilloma that was situated distalto the site of stenosis.4 The lesions may or may not bepedunculated. Moreover, most reported SEPs havebeen described as having wart-like morphology.4Treatment options are chosen based on each individualcase. The nonsurgical approach involves endoscopicexcision of the lesion using snare electrocautery oryttrium aluminum garnet (YAG) laser.7,8 This is utilisedbecause malignant spread to other areas of therespiratory tract may necessitate further removal proce-dures, so that as much pulmonary function can be savedas possible.8 The use of laser may render it impossibleto conduct a thorough pathological inspection of theentire papilloma, which might have contained evidenceof metaplastic change at the tumour base.8 Snareelectrocautery should, therefore, be the method ofchoice rather than YAG laser, because it allows for anexamination of the complete papilloma for signs ofmalignancy.7 Surgery remains the most effective andmost frequently employed method of treatment since itensures removal of any occult malignancy and preventsrecurrence that is sometimes seen after endoscopicexcision owing to incomplete tumour resection.7,8 Duringthe surgical procedure, emphasis is placed on sparingas much lung tissue as possible in order to conserve thepatient's pulmonary function.7-9 The choice of treatmentthe histologic subtype: surgical removal is the preferredapproach for squamous papillomas on account of theirknown malignant potential.1 On the other hand, forglandular papillomas (in which malignant transformationhas never been reported), endoscopic treatment isSolitary endobronchial papillomaJournal of the College of Physicians and Surgeons Pakistan 2019, Vol. 29 (Supplement 2): S120-S122 S121Figure 1: Bronchoscopy reveals a polypoid mass, measuring 3 × 5 × 4 cm,located at the origin of right upper lobe bronchus. Note the characteristic softfriable appearance of the tumour.Figure 2: Low-power view showing a papillometous lesion with centralfibrovascular cores (H&E, x 4).Muhammad Soban Arif Maan, Muhammad Waleed, Amir Humza Sohail, Asna Sulaiman and Javaid Ahmed KhanS122 Journal of the College of Physicians and Surgeons Pakistan 2019, Vol. 29 (Supplement 2): S120-S122recommended because it is less invasive.1 Ultimately,the choice of most suitable treatment hinges upon thelocation, size and histologic subtype of the lesion. It alsodependson whether the lesion is recurring every fewmonths to years after resection. Patient consent is alsoof paramount importance. Some patients, especiallythose in developing countries, may not have healthinsurance and lack the means to undergo any excisionprocedure. Such patiens are monitored carefully on aregular basis. Others might have financial means but areunwilling and apprehensive about surgery. As statedabove, our patient also denied surgery because of thisfear. Therefore, he was closely followed. When reviewedtwo months and six months later, he was stable andstated that there had been no further episodes ofhemoptysis.In a middle-aged to elderly smoker complaining of hemo-ptysis, a SEPs should be considered among thedifferentials, especially if the other more commoncauses have been ruled out. Moreover, in instanceswhere a patient declines treatment, the papilloma needsto be monitored closely at regular intervals because ofthe risk of malignant transformation. Monitoring shouldalso be continued for patients who have had thepapilloma removed.PATIENT’S CONSENT:The authors obtained written informed consent andpermission from the patient to submit this case reportand the associated images for publication.CONFLICT OF INTEREST:Authors declared no conflict of interest.AUTHORS’ CONTRIBUTION:MSAM, MW, AHS, AS, JAK: Manuscript planning, literaturesearch, writing, interpretion of available literature andderivation of relevant conclusions.REFERENCES1. Tryfon S, Dramba V, Zoglopitis F, Iakovidis D, Sakkas L,Kontakiotis T, et al. Solitary papillomas of the lower airways:Epidemiological, clinical, and therapeutic data during a 22-yearperiod and review of the literature. J Thorac Oncol 2012; 7:643-8.2. Singh SK, Tiwari KK. Etiology of hemoptysis: A retrospectivestudy from a tertiary care hospital from northern MadhyaPradesh, India. Indian J Tuberc 2016; 63:44-7.3. Feng AN, Wu HY, Zhou Q, Sun Q, Fan XS, Zhang YF, et al.Solitary endobronchial papillomas with false impression ofmalignant transformation: Report of two cases and review ofthe literature. Int J Clin Exp Pathol 2015; 8:8607-12.4. Paganin F, Prevot M, Noel JB, Frejeville M, Arvin-Berod C,Bourdin A. A solitary bronchial papilloma with unusual endo-scopic presentation: Case study and literature review. BMCPulm Med 2009; 9:40.5. Stevic R, Milenkovic B. Tracheobronchial tumors. J Thorac Dis2016; 8:3401-13.6. Ghobain MA. Solitary endobronchial papilloma with malignanttransformation and concomitant TB infection: Case report andliterature review. Case Rep Pulmonol 2017; 2017:1606432.7. Inoue Y, Oka M, Ishii H, Kimino K, Kishikawa M, Ito M, et al.A solitary bronchial papilloma with malignant changes. InternMed 2001; 40:56-60.8. McNamee CJ, Lien D, Puttagunta L, Conlan AA. Solitarysquamous papillomas of the bronchus: A case report andliterature review. J Thorac Cardiovasc Surg 2003; 126:861-3.9. Himuro N, Niiya Y, Minakata T, Oshima Y, Kataoka D, Tazawa S,et al. A solitary bronchial squamous cell papilloma withincreased 18-fluorodeoxyglucose uptake and high serumlevels of squamous cell carcinoma antigen. J Thorac Dis 2018;10:E435-E437.

Solitary endobronchial papilloma: A rare entity

https://ecommons.aku.edu/cgi/viewcontent.cgi?article=1153&amp;context=pakistan_fhs_mc_med_pulm_critcare

Solitary endobronchial papilloma: A rare entity

Abstract

Similar works

Full text

Available Versions

eCommons@AKU