Pleomorphic adenoma of the trachea: A case report and review of the literature

Table 1 Summary of presenting characteristics of tracheobronchial pleomorphic adenoma reported in the English medical literature

Ref.	Age	Sex	Clinical presentation	Course (mo)	Tumor site	Tumor size (cm)	Immunohistochemical staining	Treatment	Comorbidities	Complications	Clinical follow-up period (mo)
Heifetz et al[18], 1992	15	M	Asthma, wheezing, and dyspnea	12	Upper trachea (level of the fourth ring)	2.5 × 2.5 × 2.5	+: CK AE1/3, S-100, actin, vimentin, EMA, GFAP	CO2 laser bronchoscopy	No	No	Alive with no evidence of recurrence (6)
Basaklar et al[19], 1994	11	F	Nonproductive harsh cough, high fever, nausea, vomiting, and night sweats	1.5	Right upper lobe bronchus	2	Not available	Surgical resection	Atelectasis, multiple mediastinal and peribronchial lymphadenopathies	No	Not available
Sweeney et al[20], 1996	27	M	Incidental (asymptomatic)	Not available	Right lower lobe bronchus	3 × 5	+: CK, EMA, S 100, SMA	A lower lobectomy	No	No	Not available
Paik et al[21], 1996	50	M	Mild dyspnea upon exertion	3	Mid trachea (4 cm above the carina)	2 × 2	Not available	Right thoracotomy with segmental resection and end-to-end anastomosis	No	No	Alive with no evidence of recurrence (18 d)
Bizal et al[22], 1997	27	M	Dyspnea upon exertion and intermittent wheezing	12	Lower trachea (2 cm above the carina)	2.5	Not available	Surgical resection and primary anastonosis performed through right thoracotomy	No	No	Alive with no evidence of recurrence (6)
Paik et al[23], 1997	48	F	Dyspnea upon exertion and productive cough with wheezing	3	Lower trachea	1.5 × 1.2	+: Vimentin, CK, S-100, GFAP, SMA	Tracheal wedge resection	No	No	Not available
Pomp et al[24], 1998	79	F	Increasing stridor, dyspnea and a dry cough	2	Upper trachea (level of fifth ring)	2	Not available	Radiotherapy, excision through rigid bronchoscopy	No	Recurrent PA of the trachea	Not available
Pomp et al[24], 1998	58	F	Increasing dyspnea and stridor	6	Upper trachea (below the larynx)	90% occlusion	Not available	Excision via tracheotomy	No	No	Alive with no evidence of recurrence (12)
Kim et al[25], 2000	15	M	Asthma, dyspnea and stridor	5	Upper trachea	1.5	Not available	Segmental tracheal resection and end-to-end anastomosis	No	No	Alive with no evidence of recurrence (12)
Baghai-Wadji et al[7], 2006	8	M	Asthma, fever, productive cough, severe wheezing, and respiratory distress	10 d	Lower trachea	90% occlusion	+: Chromogranin, NSE, CK	Surgical resection and tracheal reconstruction (pericardial patch graft)	Pneumonia	No	Alive with no evidence of recurrence (6)
Aribas et al[8], 2007	42	F	Asthma, severe dyspnea	2 yr	Lower trachea	2 × 2	+: Vimentin, GFAP, S-100	Segmental tracheal resection and end-to-end anastomosis	No	Tracheal stenosis	Alive with no evidence of recurrence (5 yr)
Ashwaq et al[26], 2007	37	M	Spontaneous hemoptysis	8	Mid trachea	2 × 2	Not available	Excision with cold instrument via suspension laryngoscopy	No	No	Alive with no evidence of recurrence (3)
Matsubara et al[27], 2008	71	M	Incidental (asymptomatic)	Not available	Left main bronchus	Not available	+: polyclonal anti-S-100, anti-GFAP	Endoscopic resection with electrosurgical snaring and APC	No	No	Alive with no evidence of recurrence (6)
Fitchett et al[11], 2008	65	M	Hoarse barking cough	5	Right main bronchus	1.3	Not available	Endoscopic resection with diathermy snare	No	No	Not available
Kamiyoshihara et al[28], 2009	34	F	Dyspnea upon exertion	3	Left main bronchus	1.2 × 1.1	Not available	Surgical resection with wedge bronchiectomy	No	No	Alive with no evidence of recurrence (11)
Tanaka et al[13], 2010	57	F	A neck mass	10 yr	Right lobe of the thyroid (originating from the trachea)	3.25 × 2.09	+: SMA, 34bE12; -: P53 and ki67	Surgical resection and direct anastomosis	No	No	Not available
Kajikawa et al[9], 2010	55	M	Asthma, dyspnea with wheezing	2 yr	Lower trachea	Not available	Not available	Endoscopic resection with APC, electrocautery and rigid bronchoscopic coring	No	No	Alive with no evidence of recurrence (7)
Lin et al[29], 2011	36	F	Bronchial asthma, worsening shortness of breath	6	Lower trachea(3 cm above the carina)	2 × 2 × 2	Not available	Segmental tracheal resection and anastomosis	Allergic rhinitis	No	Not available
Goto et al[30], 2011	71	M	Progressive dyspnea	Not available	Left main bronchus	2.5 × 2	+: CK AE1/3, SMA	Endoscopic resection with electrosurgical snaring	Chronic obstructive pulmonary disease, squamous cell; carcinoma (pT2N0M0, stage IB)	No	Alive with no evidence of recurrence (2)
Solak et al[15], 2012	46	F	Severe dyspnea	12	Upper trachea	3 × 2	Not available	Collar incision with partial sternotomy and end-to-end anastomosis	No	No	Alive with no evidence of recurrence (1)
Park et al[16], 2013	59	M	Dyspnea upon exertion	3	Mid trachea	2 × 2	+: CK, CK 19, EMA, S100, p63	Right thoracotomy with segmental resection and end-to-end anastomosis	Active pulmonary tuberculosis	No	Alive with no evidence of recurrence (5 yr)
Lee et al[31], 2014	54	F	Blunt chest pain upon bending forward	2 wk	Posterior mediastinum (originating from the left main bronchus)	6.0 × 4.5 × 2.5	+: P63 and SMA	Video-assisted thoracic surgery	No	No	Alive with no evidence of recurrence (2 yr)
Casillas-Enríquez et al[32], 2014	33	F	Productive cough, wheezing, and occasional hemoptysis	4 yr	Upper trachea	80% occlusion	Not available	Endoscopic resection with APC	No	No	Alive with no evidence of recurrence (8)
Sim et al[33], 2014	32	F	Dyspnea upon exertion and chronic cough with wheezing	8	Lower trachea	1.8 × 1.6	Not available	Endoscopic resection with rigid forceps and APC	Situs inversus	No	Alive with no evidence of recurrence (1)
Zhu et al[3], 2018	38	F	Progressive shortness of breath	5 yr	Right main bronchus	1.42 × 0.96	Not available	Endoscopic resection with electrosurgical snare and APC	No	No	Alive with no evidence of recurrence (3)
Kim et al[4], 2018	49	M	Exacerbation of dyspnea upon exertion, cough and sputum	3	Lower trachea	1.5 × 1.3 × 1.3	+: CK 5/6, CK, p53	Right thoracotomy with segmental resection and anastomosis with tracheobronchoplasty	Active pulmonary tuberculoma	No	Alive with no evidence of recurrence (3)
David et al[5], 2020	83	F	Worsening shortness of breath and waking up with blood in her oropharynx	1	Upper trachea (3.0 cm below the vocal fold edge)	1.6 × 1.3	+: P63, SMA; -: Chromogranin, synaptophysin	Endoscopic excision with fiber-based CO2 laser and rigid bronchoscope	Hypertension, rheumatoid arthritis	No	Not available
Takahashi et al[6], 2019	51	F	Asthma, cough and wheezing at night	2	Upper trachea (periphery 30 mm from the glottis)	1.5	Not available	Endoscopic resection with electrosurgical snaring and forceps	No	No	Alive with no evidence of recurrence (30)
Our case	49	F	Dyspnea upon exertion and chronic cough with wheezing	2 yr	Lower trachea	2.4 × 2.1	+ ：CK, CK 5/6, p63, S-100, Ki-67 (10%); - ：TTF-1, CK 7	Endoscopic resection electrosurgical snare, cryotherapy and APC	No	No	Alive with no evidence of recurrence (3)

CK: Cytokeratin; EMA: Epithelial membrane antigen; GFAP: Glial fibrillary acidic protein; SMA: Smooth muscle actin; NSE: Neuron-specific enolase; APC: Argon plasma coagulation; TTF-1: Thyroid transcription factor-1; M: Male; F: Female; CK 7: Cytokeratin 7.