Abstracts
Tracheal diverticulum, defined as a benign outpouching of the tracheal wall, is rarely diagnosed in clinical practice. It can be congenital or acquired in origin, and most cases are asymptomatic, typically being diagnosed postmortem. We report a case of a 69-year-old woman who was hospitalized after presenting with fever, fatigue, pleuritic chest pain, and a right neck mass complicated by dysphagia. Her medical history was significant: pulmonary emphysema (alpha-1 antitrypsin deficiency); bronchiectasis; and thyroidectomy. On physical examination, she presented diminished breath sounds and muffled heart sounds, with a systolic murmur. Laboratory tests revealed elevated inflammatory markers, a CT scan showed an air-filled, multilocular mass in the right tracheal wall, and magnetic resonance imaging confirmed the CT findings. Fiberoptic bronchoscopy failed to reveal any abnormalities. Nevertheless, the patient was diagnosed with tracheal diverticulum. The treatment approach was conservative, consisting mainly of antibiotics. After showing clinical improvement, the patient was discharged.
Tracheal diseases; Pulmonary emphysema; Diverticulum; alpha 1-antitrypsin deficiency
Divertículos da traqueia são evaginações benignas da parede traqueal e raramente diagnosticados na prática clínica. Podem ser congênitos ou adquiridos, e na maioria dos casos são assintomáticos, sendo tipicamente diagnosticados em estudos post-mortem. Relatamos o caso de uma mulher de 69 anos que foi hospitalizada após apresentar febre, fadiga, dor torácica pleurítica e uma massa cervical à direita complicada por disfagia. Tinha antecedentes pessoais de enfisema pulmonar (deficiência de alfa-1 antitripsina), bronquiectasias e tireoidectomia. Ao exame físico apresentava murmúrio vesicular diminuído, hipofonese cardíaca e um sopro sistólico. Laboratorialmente apresentava marcadores inflamatórios elevados, e uma TC mostrou uma massa aérea, multiloculada na parede direita da traqueia, achados confirmados por ressonância magnética nuclear. Realizou ainda uma fibrobroncoscopia que se revelou normal. Assumiu-se o diagnóstico de divertículo da traqueia. O tratamento proposto foi conservador, consistindo principalmente de antibioticoterapia. Após melhora clínica, a paciente recebeu alta.
Doenças da traqueia; Enfisema pulmonar; Divertículo; Deficiência de alfa 1-antitripsina
Introduction
A tracheal diverticulum is a benign outpouching of the tracheal wall that is rarely
diagnosed in routine clinical practice, and there are few reports of cases in the
literature. Tracheal diverticula can be single or multiple(
11. Monroe MM, McLean M, Gross ND. Infected tracheal diverticulum
presenting as a cervical abscess Laryngoscope.
2013;123(7):1583-7.http://dx.doi.org/10.1002/lary.23978
http://dx.doi.org/10.1002/lary.23978...
2. Smelley CL, Bowen R, Nathan CO. Intermittently symptomatic tracheal
diverticulum: a case of a rare clinical phenomenon. Ear Nose Throat J.
2011;90(9):E10-2.
3. Soto-Hurtado EJ, Pe-uela-Ruíz L, Rivera-Sánchez I, Torres-Jiménez J.
Tracheal diverticulum: a review of the literature. Lung.
2006;184(6):303-7.http://dx.doi.org/10.1007/s00408-006-0010-7
http://dx.doi.org/10.1007/s00408-006-001...
4. Waite S, Sharma A, Machnicki S. Right paratracheal air cyst/tracheal
diverticulum.Appl Radiol. 2003;32(8):
5. Shah M, Joshi JM. Tracheal Diverticulum. Indian J Chest Dis Allied
Sci. 2012;54(1):39-40.
6. Han S, Dikmen E, Aydin S, Yapakci O. Tracheal diverticulum: a rare
case of dysphagia. Eur J Cardiothorac Surg.
2008;34(4):916-7http://dx.doi.org/10.1016/j.ejcts.2008.06.022
http://dx.doi.org/10.1016/j.ejcts.2008.0...
7. Haghi Z, Towhidi M, Fattahi H, Lari SM. Right paratracheal air cyst
(tracheal diverticulum). Respir Care. 2009;54(10):1409-11.
8. Sharma BG. Tracheal diverticulum: a report of 4 cases. Ear Nose
Throat J. 2009;88(1):E11.
-
99. Ortega HA, Vega NA, Comparini J, Carmo LV, Pereira, LP. Doença
cística traqueobrônquica. J Pneumol.
2001;27(6):349-54.http://dx.doi.org/10.1590/S0102-35862001000600010
http://dx.doi.org/10.1590/S0102-35862001...
) and can present either as ovoid pedunculated cysts or sessile formations,
their longest axis measuring up to 3 cm, communicating with the trachea by
channels.(
99. Ortega HA, Vega NA, Comparini J, Carmo LV, Pereira, LP. Doença
cística traqueobrônquica. J Pneumol.
2001;27(6):349-54.http://dx.doi.org/10.1590/S0102-35862001000600010
http://dx.doi.org/10.1590/S0102-35862001...
) Concerning etiology, tracheal diverticula can represent congenital
anomalies of the tracheobronchial tree or acquired outpouchings from a weakened tracheal
wall,(
11. Monroe MM, McLean M, Gross ND. Infected tracheal diverticulum
presenting as a cervical abscess Laryngoscope.
2013;123(7):1583-7.http://dx.doi.org/10.1002/lary.23978
http://dx.doi.org/10.1002/lary.23978...
,
22. Smelley CL, Bowen R, Nathan CO. Intermittently symptomatic tracheal
diverticulum: a case of a rare clinical phenomenon. Ear Nose Throat J.
2011;90(9):E10-2.
,
44. Waite S, Sharma A, Machnicki S. Right paratracheal air cyst/tracheal
diverticulum.Appl Radiol. 2003;32(8):
,
55. Shah M, Joshi JM. Tracheal Diverticulum. Indian J Chest Dis Allied
Sci. 2012;54(1):39-40.
,
88. Sharma BG. Tracheal diverticulum: a report of 4 cases. Ear Nose
Throat J. 2009;88(1):E11.
9. Ortega HA, Vega NA, Comparini J, Carmo LV, Pereira, LP. Doença
cística traqueobrônquica. J Pneumol.
2001;27(6):349-54.http://dx.doi.org/10.1590/S0102-35862001000600010
http://dx.doi.org/10.1590/S0102-35862001...
-
1010. Charest M, Sirois C, Carter Y, Rousseau J. Infected tracheal
diverticulum mimicking an aggressive mediastinal lesion on FDG PET/CT: an interesting
case with review of the literature. Br J Radiol.
2012;85(1009):e17-21.http://dx.doi.org/10.1259/bjr/32814390
http://dx.doi.org/10.1259/bjr/32814390...
) the two conditions differing mainly in relation to the site of implantation
and the histological features of the wall.(
33. Soto-Hurtado EJ, Pe-uela-Ruíz L, Rivera-Sánchez I, Torres-Jiménez J.
Tracheal diverticulum: a review of the literature. Lung.
2006;184(6):303-7.http://dx.doi.org/10.1007/s00408-006-0010-7
http://dx.doi.org/10.1007/s00408-006-001...
,
66. Han S, Dikmen E, Aydin S, Yapakci O. Tracheal diverticulum: a rare
case of dysphagia. Eur J Cardiothorac Surg.
2008;34(4):916-7http://dx.doi.org/10.1016/j.ejcts.2008.06.022
http://dx.doi.org/10.1016/j.ejcts.2008.0...
,
1111. Kokkonouzis I, Haramis D, Kornezos I, Moschouris H, Katsenos S,
Bouchara S. Tracheal diverticulum in an asymptomatic male: a case report. Cases J.
2008;1(1):181. http://dx.doi.org/10.1186/1757-1626-1-181
http://dx.doi.org/10.1186/1757-1626-1-18...
) Congenital tracheal diverticula are usually smaller and feature a narrower
communication with the trachea than do those that are acquired. (
11. Monroe MM, McLean M, Gross ND. Infected tracheal diverticulum
presenting as a cervical abscess Laryngoscope.
2013;123(7):1583-7.http://dx.doi.org/10.1002/lary.23978
http://dx.doi.org/10.1002/lary.23978...
2. Smelley CL, Bowen R, Nathan CO. Intermittently symptomatic tracheal
diverticulum: a case of a rare clinical phenomenon. Ear Nose Throat J.
2011;90(9):E10-2.
-
33. Soto-Hurtado EJ, Pe-uela-Ruíz L, Rivera-Sánchez I, Torres-Jiménez J.
Tracheal diverticulum: a review of the literature. Lung.
2006;184(6):303-7.http://dx.doi.org/10.1007/s00408-006-0010-7
http://dx.doi.org/10.1007/s00408-006-001...
,
55. Shah M, Joshi JM. Tracheal Diverticulum. Indian J Chest Dis Allied
Sci. 2012;54(1):39-40.
,
88. Sharma BG. Tracheal diverticulum: a report of 4 cases. Ear Nose
Throat J. 2009;88(1):E11.
,
99. Ortega HA, Vega NA, Comparini J, Carmo LV, Pereira, LP. Doença
cística traqueobrônquica. J Pneumol.
2001;27(6):349-54.http://dx.doi.org/10.1590/S0102-35862001000600010
http://dx.doi.org/10.1590/S0102-35862001...
,
1111. Kokkonouzis I, Haramis D, Kornezos I, Moschouris H, Katsenos S,
Bouchara S. Tracheal diverticulum in an asymptomatic male: a case report. Cases J.
2008;1(1):181. http://dx.doi.org/10.1186/1757-1626-1-181
http://dx.doi.org/10.1186/1757-1626-1-18...
12. Danielson GP, Jedlovszky V, Landigran GP. Tracheal diverticulum: a
rare finding in a patient with worsening chronic cough. Ear Nose Throat J.
2008;87(8):474-5.
-
1313. Agustí EB, Vecina VM, Figuerola CR, Gaspà MM. Tracheal diverticulum:
a case report. Acta Otorrinolaringol Esp.
2007;58(6):278-9.http://dx.doi.org/10.1016/S2173-5735(07)70349-6
http://dx.doi.org/10.1016/S2173-5735(07)...
) The former are frequently right-sided, occurring 4-5 cm below the vocal
cords or a few centimeters above the carina,(
33. Soto-Hurtado EJ, Pe-uela-Ruíz L, Rivera-Sánchez I, Torres-Jiménez J.
Tracheal diverticulum: a review of the literature. Lung.
2006;184(6):303-7.http://dx.doi.org/10.1007/s00408-006-0010-7
http://dx.doi.org/10.1007/s00408-006-001...
,
44. Waite S, Sharma A, Machnicki S. Right paratracheal air cyst/tracheal
diverticulum.Appl Radiol. 2003;32(8):
,
1313. Agustí EB, Vecina VM, Figuerola CR, Gaspà MM. Tracheal diverticulum:
a case report. Acta Otorrinolaringol Esp.
2007;58(6):278-9.http://dx.doi.org/10.1016/S2173-5735(07)70349-6
http://dx.doi.org/10.1016/S2173-5735(07)...
14. Pinot D, Breen D, Peloni JM, Gaubert JY, Dutau H, Vervloet D. An
incidental finding in a 34-year-old male under investigation for haemoptysis.
Diagnosis: The radiological and endoscopic images demonstrate a complex defect along
the posterior tracheal wall consistent with acquired tracheal diverticulum. Eur
Respir J. 2009;33(5):1227-9. http://dx.doi.org/10.1183/09031936.00131908
http://dx.doi.org/10.1183/09031936.00131...
-
1515. Elicora A, Liman ST, Topcu S, Akgul AG. Congenital tracheal
diverticulum seen in adult age. Eur J Cardiothorac Surg.
2012;41(6):1399.http://dx.doi.org/10.1093/ejcts/ezr264
http://dx.doi.org/10.1093/ejcts/ezr264...
) and are histologically similar to the tracheal wall, comprising respiratory
epithelia, smooth muscle, and cartilage, whereas acquired tracheal diverticula are
composed exclusively of respiratory epithelia.(
33. Soto-Hurtado EJ, Pe-uela-Ruíz L, Rivera-Sánchez I, Torres-Jiménez J.
Tracheal diverticulum: a review of the literature. Lung.
2006;184(6):303-7.http://dx.doi.org/10.1007/s00408-006-0010-7
http://dx.doi.org/10.1007/s00408-006-001...
,
1111. Kokkonouzis I, Haramis D, Kornezos I, Moschouris H, Katsenos S,
Bouchara S. Tracheal diverticulum in an asymptomatic male: a case report. Cases J.
2008;1(1):181. http://dx.doi.org/10.1186/1757-1626-1-181
http://dx.doi.org/10.1186/1757-1626-1-18...
) The lack of cartilage in the acquired form is thought to be due to its
origin, because it is caused by increased intra-luminal pressure or by the weakening of
the structures after surgical procedures,(
1010. Charest M, Sirois C, Carter Y, Rousseau J. Infected tracheal
diverticulum mimicking an aggressive mediastinal lesion on FDG PET/CT: an interesting
case with review of the literature. Br J Radiol.
2012;85(1009):e17-21.http://dx.doi.org/10.1259/bjr/32814390
http://dx.doi.org/10.1259/bjr/32814390...
,
1313. Agustí EB, Vecina VM, Figuerola CR, Gaspà MM. Tracheal diverticulum:
a case report. Acta Otorrinolaringol Esp.
2007;58(6):278-9.http://dx.doi.org/10.1016/S2173-5735(07)70349-6
http://dx.doi.org/10.1016/S2173-5735(07)...
) resulting in evagination of the mucous membrane through vulnerable points
in the trachea.(
22. Smelley CL, Bowen R, Nathan CO. Intermittently symptomatic tracheal
diverticulum: a case of a rare clinical phenomenon. Ear Nose Throat J.
2011;90(9):E10-2.
,
33. Soto-Hurtado EJ, Pe-uela-Ruíz L, Rivera-Sánchez I, Torres-Jiménez J.
Tracheal diverticulum: a review of the literature. Lung.
2006;184(6):303-7.http://dx.doi.org/10.1007/s00408-006-0010-7
http://dx.doi.org/10.1007/s00408-006-001...
,
99. Ortega HA, Vega NA, Comparini J, Carmo LV, Pereira, LP. Doença
cística traqueobrônquica. J Pneumol.
2001;27(6):349-54.http://dx.doi.org/10.1590/S0102-35862001000600010
http://dx.doi.org/10.1590/S0102-35862001...
,
1111. Kokkonouzis I, Haramis D, Kornezos I, Moschouris H, Katsenos S,
Bouchara S. Tracheal diverticulum in an asymptomatic male: a case report. Cases J.
2008;1(1):181. http://dx.doi.org/10.1186/1757-1626-1-181
http://dx.doi.org/10.1186/1757-1626-1-18...
,
1212. Danielson GP, Jedlovszky V, Landigran GP. Tracheal diverticulum: a
rare finding in a patient with worsening chronic cough. Ear Nose Throat J.
2008;87(8):474-5.
) Therefore, acquired tracheal diverticula can appear at any level, although
they typically occur along the right posterolateral wall(
66. Han S, Dikmen E, Aydin S, Yapakci O. Tracheal diverticulum: a rare
case of dysphagia. Eur J Cardiothorac Surg.
2008;34(4):916-7http://dx.doi.org/10.1016/j.ejcts.2008.06.022
http://dx.doi.org/10.1016/j.ejcts.2008.0...
,
1616. Rahalkar MD, Lakhkar DL, Joshi SW, Gundawar S. Tracheal
diverticula-report of 2 cases. Indian J Radiol Imaging.
2004;14(2):197-8.
,
1717. Soto PC, Congregado M, Loscertales J. Divertículo traqueal adquirido
como causa de intubación orotracheal complicada. Arch Bronconeumol. 2012;48(2):64-6.
http://dx.doi.org/10.1016/j.arbres.2011.09.006
http://dx.doi.org/10.1016/j.arbres.2011....
) near the thoracic inlet.(
11. Monroe MM, McLean M, Gross ND. Infected tracheal diverticulum
presenting as a cervical abscess Laryngoscope.
2013;123(7):1583-7.http://dx.doi.org/10.1002/lary.23978
http://dx.doi.org/10.1002/lary.23978...
,
22. Smelley CL, Bowen R, Nathan CO. Intermittently symptomatic tracheal
diverticulum: a case of a rare clinical phenomenon. Ear Nose Throat J.
2011;90(9):E10-2.
,
88. Sharma BG. Tracheal diverticulum: a report of 4 cases. Ear Nose
Throat J. 2009;88(1):E11.
)
Tracheal diverticula are usually asymptomatic, and their frequency can therefore be
underestimated.(
55. Shah M, Joshi JM. Tracheal Diverticulum. Indian J Chest Dis Allied
Sci. 2012;54(1):39-40.
,
77. Haghi Z, Towhidi M, Fattahi H, Lari SM. Right paratracheal air cyst
(tracheal diverticulum). Respir Care. 2009;54(10):1409-11.
,
1818. Djamouri F, Le Pimpec Barthes F, Pheulpin G, Grignet JP, Carnot F,
Riquet M Air-filled cysts of tracheal origin: nosological problems and actual
frequency [Article in French]. Rev Mal Respir. 2002;19(4):523-6.
) In most cases, the diagnosis is made on the basis of features observed on
X-rays, on CT scans,(
77. Haghi Z, Towhidi M, Fattahi H, Lari SM. Right paratracheal air cyst
(tracheal diverticulum). Respir Care. 2009;54(10):1409-11.
,
1616. Rahalkar MD, Lakhkar DL, Joshi SW, Gundawar S. Tracheal
diverticula-report of 2 cases. Indian J Radiol Imaging.
2004;14(2):197-8.
,
1717. Soto PC, Congregado M, Loscertales J. Divertículo traqueal adquirido
como causa de intubación orotracheal complicada. Arch Bronconeumol. 2012;48(2):64-6.
http://dx.doi.org/10.1016/j.arbres.2011.09.006
http://dx.doi.org/10.1016/j.arbres.2011....
) during bronchoscopy,(
1313. Agustí EB, Vecina VM, Figuerola CR, Gaspà MM. Tracheal diverticulum:
a case report. Acta Otorrinolaringol Esp.
2007;58(6):278-9.http://dx.doi.org/10.1016/S2173-5735(07)70349-6
http://dx.doi.org/10.1016/S2173-5735(07)...
,
1616. Rahalkar MD, Lakhkar DL, Joshi SW, Gundawar S. Tracheal
diverticula-report of 2 cases. Indian J Radiol Imaging.
2004;14(2):197-8.
) or in autopsies.(
11. Monroe MM, McLean M, Gross ND. Infected tracheal diverticulum
presenting as a cervical abscess Laryngoscope.
2013;123(7):1583-7.http://dx.doi.org/10.1002/lary.23978
http://dx.doi.org/10.1002/lary.23978...
2. Smelley CL, Bowen R, Nathan CO. Intermittently symptomatic tracheal
diverticulum: a case of a rare clinical phenomenon. Ear Nose Throat J.
2011;90(9):E10-2.
-
33. Soto-Hurtado EJ, Pe-uela-Ruíz L, Rivera-Sánchez I, Torres-Jiménez J.
Tracheal diverticulum: a review of the literature. Lung.
2006;184(6):303-7.http://dx.doi.org/10.1007/s00408-006-0010-7
http://dx.doi.org/10.1007/s00408-006-001...
,
55. Shah M, Joshi JM. Tracheal Diverticulum. Indian J Chest Dis Allied
Sci. 2012;54(1):39-40.
6. Han S, Dikmen E, Aydin S, Yapakci O. Tracheal diverticulum: a rare
case of dysphagia. Eur J Cardiothorac Surg.
2008;34(4):916-7http://dx.doi.org/10.1016/j.ejcts.2008.06.022
http://dx.doi.org/10.1016/j.ejcts.2008.0...
7. Haghi Z, Towhidi M, Fattahi H, Lari SM. Right paratracheal air cyst
(tracheal diverticulum). Respir Care. 2009;54(10):1409-11.
-
88. Sharma BG. Tracheal diverticulum: a report of 4 cases. Ear Nose
Throat J. 2009;88(1):E11.
,
1111. Kokkonouzis I, Haramis D, Kornezos I, Moschouris H, Katsenos S,
Bouchara S. Tracheal diverticulum in an asymptomatic male: a case report. Cases J.
2008;1(1):181. http://dx.doi.org/10.1186/1757-1626-1-181
http://dx.doi.org/10.1186/1757-1626-1-18...
,
1212. Danielson GP, Jedlovszky V, Landigran GP. Tracheal diverticulum: a
rare finding in a patient with worsening chronic cough. Ear Nose Throat J.
2008;87(8):474-5.
,
1919. Teh BM, Hall C, Kleid S. Infected tracheocoele (acquired tracheal
diverticulum): case report and literature review. J Laryngol Otol.
2011;125(5):540-5.http://dx.doi.org/10.1017/S0022215110003026
http://dx.doi.org/10.1017/S0022215110003...
) Consequently, the reported incidence varies depending on the diagnostic
tool used. For example, in a postmortem study, the incidence was estimated to be
approximately 1%,(
11. Monroe MM, McLean M, Gross ND. Infected tracheal diverticulum
presenting as a cervical abscess Laryngoscope.
2013;123(7):1583-7.http://dx.doi.org/10.1002/lary.23978
http://dx.doi.org/10.1002/lary.23978...
,
44. Waite S, Sharma A, Machnicki S. Right paratracheal air cyst/tracheal
diverticulum.Appl Radiol. 2003;32(8):
,
66. Han S, Dikmen E, Aydin S, Yapakci O. Tracheal diverticulum: a rare
case of dysphagia. Eur J Cardiothorac Surg.
2008;34(4):916-7http://dx.doi.org/10.1016/j.ejcts.2008.06.022
http://dx.doi.org/10.1016/j.ejcts.2008.0...
7. Haghi Z, Towhidi M, Fattahi H, Lari SM. Right paratracheal air cyst
(tracheal diverticulum). Respir Care. 2009;54(10):1409-11.
8. Sharma BG. Tracheal diverticulum: a report of 4 cases. Ear Nose
Throat J. 2009;88(1):E11.
-
99. Ortega HA, Vega NA, Comparini J, Carmo LV, Pereira, LP. Doença
cística traqueobrônquica. J Pneumol.
2001;27(6):349-54.http://dx.doi.org/10.1590/S0102-35862001000600010
http://dx.doi.org/10.1590/S0102-35862001...
,
1111. Kokkonouzis I, Haramis D, Kornezos I, Moschouris H, Katsenos S,
Bouchara S. Tracheal diverticulum in an asymptomatic male: a case report. Cases J.
2008;1(1):181. http://dx.doi.org/10.1186/1757-1626-1-181
http://dx.doi.org/10.1186/1757-1626-1-18...
,
1313. Agustí EB, Vecina VM, Figuerola CR, Gaspà MM. Tracheal diverticulum:
a case report. Acta Otorrinolaringol Esp.
2007;58(6):278-9.http://dx.doi.org/10.1016/S2173-5735(07)70349-6
http://dx.doi.org/10.1016/S2173-5735(07)...
,
1414. Pinot D, Breen D, Peloni JM, Gaubert JY, Dutau H, Vervloet D. An
incidental finding in a 34-year-old male under investigation for haemoptysis.
Diagnosis: The radiological and endoscopic images demonstrate a complex defect along
the posterior tracheal wall consistent with acquired tracheal diverticulum. Eur
Respir J. 2009;33(5):1227-9. http://dx.doi.org/10.1183/09031936.00131908
http://dx.doi.org/10.1183/09031936.00131...
) compared with 0.3% in children diagnosed through bronchoscopy.(
77. Haghi Z, Towhidi M, Fattahi H, Lari SM. Right paratracheal air cyst
(tracheal diverticulum). Respir Care. 2009;54(10):1409-11.
,
88. Sharma BG. Tracheal diverticulum: a report of 4 cases. Ear Nose
Throat J. 2009;88(1):E11.
,
1414. Pinot D, Breen D, Peloni JM, Gaubert JY, Dutau H, Vervloet D. An
incidental finding in a 34-year-old male under investigation for haemoptysis.
Diagnosis: The radiological and endoscopic images demonstrate a complex defect along
the posterior tracheal wall consistent with acquired tracheal diverticulum. Eur
Respir J. 2009;33(5):1227-9. http://dx.doi.org/10.1183/09031936.00131908
http://dx.doi.org/10.1183/09031936.00131...
) However, in a study employing cervical CT, Buterbaugh & Erly estimated
that paratracheal air cyst (i.e., tracheal diverticulum) occurs in approximately 3.7% of
the population.(
2020. Buterbaugh JE, Erly WK. Paratracheal air cysts: a common finding on
routine CT Examinations of the cervical spine and neck that may mimic
pneumomediastinum in patients with traumatic injuries. AJNR Am J Neuroradiol.
2008;29(6):1218-21. http://dx.doi.org/10.3174/ajnr.A1058
http://dx.doi.org/10.3174/ajnr.A1058...
)
Although infrequent and nonspecific, symptoms can develop in individuals with tracheal
diverticulum.(
33. Soto-Hurtado EJ, Pe-uela-Ruíz L, Rivera-Sánchez I, Torres-Jiménez J.
Tracheal diverticulum: a review of the literature. Lung.
2006;184(6):303-7.http://dx.doi.org/10.1007/s00408-006-0010-7
http://dx.doi.org/10.1007/s00408-006-001...
) Because the diverticula usually act as reservoirs for respiratory
secretions, they are sometimes associated with chronic cough and can become
infected.(
22. Smelley CL, Bowen R, Nathan CO. Intermittently symptomatic tracheal
diverticulum: a case of a rare clinical phenomenon. Ear Nose Throat J.
2011;90(9):E10-2.
3. Soto-Hurtado EJ, Pe-uela-Ruíz L, Rivera-Sánchez I, Torres-Jiménez J.
Tracheal diverticulum: a review of the literature. Lung.
2006;184(6):303-7.http://dx.doi.org/10.1007/s00408-006-0010-7
http://dx.doi.org/10.1007/s00408-006-001...
4. Waite S, Sharma A, Machnicki S. Right paratracheal air cyst/tracheal
diverticulum.Appl Radiol. 2003;32(8):
-
55. Shah M, Joshi JM. Tracheal Diverticulum. Indian J Chest Dis Allied
Sci. 2012;54(1):39-40.
,
88. Sharma BG. Tracheal diverticulum: a report of 4 cases. Ear Nose
Throat J. 2009;88(1):E11.
,
99. Ortega HA, Vega NA, Comparini J, Carmo LV, Pereira, LP. Doença
cística traqueobrônquica. J Pneumol.
2001;27(6):349-54.http://dx.doi.org/10.1590/S0102-35862001000600010
http://dx.doi.org/10.1590/S0102-35862001...
,
1111. Kokkonouzis I, Haramis D, Kornezos I, Moschouris H, Katsenos S,
Bouchara S. Tracheal diverticulum in an asymptomatic male: a case report. Cases J.
2008;1(1):181. http://dx.doi.org/10.1186/1757-1626-1-181
http://dx.doi.org/10.1186/1757-1626-1-18...
,
1414. Pinot D, Breen D, Peloni JM, Gaubert JY, Dutau H, Vervloet D. An
incidental finding in a 34-year-old male under investigation for haemoptysis.
Diagnosis: The radiological and endoscopic images demonstrate a complex defect along
the posterior tracheal wall consistent with acquired tracheal diverticulum. Eur
Respir J. 2009;33(5):1227-9. http://dx.doi.org/10.1183/09031936.00131908
http://dx.doi.org/10.1183/09031936.00131...
) Although less common, dyspnea, dysphagia, dysphonia, recurrent nerve
paralysis, cervical neck swelling, hematemesis, and hemoptysis have been described in
patients with tracheal diverticula.(
22. Smelley CL, Bowen R, Nathan CO. Intermittently symptomatic tracheal
diverticulum: a case of a rare clinical phenomenon. Ear Nose Throat J.
2011;90(9):E10-2.
,
88. Sharma BG. Tracheal diverticulum: a report of 4 cases. Ear Nose
Throat J. 2009;88(1):E11.
,
1212. Danielson GP, Jedlovszky V, Landigran GP. Tracheal diverticulum: a
rare finding in a patient with worsening chronic cough. Ear Nose Throat J.
2008;87(8):474-5.
) Tracheal diverticulum can also be associated with abnormal pulmonary
function and obstructive lung disease,(
77. Haghi Z, Towhidi M, Fattahi H, Lari SM. Right paratracheal air cyst
(tracheal diverticulum). Respir Care. 2009;54(10):1409-11.
8. Sharma BG. Tracheal diverticulum: a report of 4 cases. Ear Nose
Throat J. 2009;88(1):E11.
-
99. Ortega HA, Vega NA, Comparini J, Carmo LV, Pereira, LP. Doença
cística traqueobrônquica. J Pneumol.
2001;27(6):349-54.http://dx.doi.org/10.1590/S0102-35862001000600010
http://dx.doi.org/10.1590/S0102-35862001...
) mainly emphysema,(
44. Waite S, Sharma A, Machnicki S. Right paratracheal air cyst/tracheal
diverticulum.Appl Radiol. 2003;32(8):
,
2020. Buterbaugh JE, Erly WK. Paratracheal air cysts: a common finding on
routine CT Examinations of the cervical spine and neck that may mimic
pneumomediastinum in patients with traumatic injuries. AJNR Am J Neuroradiol.
2008;29(6):1218-21. http://dx.doi.org/10.3174/ajnr.A1058
http://dx.doi.org/10.3174/ajnr.A1058...
) although no relationship between tracheal diverticulum and
alpha-1-antitrypsin deficiency has been described to date.
In establishing a diagnosis of tracheal diverticulum, CT plays a fundamental role,
providing information concerning the location, origin, and size of the lesion, thus
helping distinguish between the congenital and acquired forms. Fiberoptic bronchoscopy,
although useful, can yield false-negative results, because it often misses the
communication point with the trachea. (
33. Soto-Hurtado EJ, Pe-uela-Ruíz L, Rivera-Sánchez I, Torres-Jiménez J.
Tracheal diverticulum: a review of the literature. Lung.
2006;184(6):303-7.http://dx.doi.org/10.1007/s00408-006-0010-7
http://dx.doi.org/10.1007/s00408-006-001...
,
44. Waite S, Sharma A, Machnicki S. Right paratracheal air cyst/tracheal
diverticulum.Appl Radiol. 2003;32(8):
,
66. Han S, Dikmen E, Aydin S, Yapakci O. Tracheal diverticulum: a rare
case of dysphagia. Eur J Cardiothorac Surg.
2008;34(4):916-7http://dx.doi.org/10.1016/j.ejcts.2008.06.022
http://dx.doi.org/10.1016/j.ejcts.2008.0...
,
1010. Charest M, Sirois C, Carter Y, Rousseau J. Infected tracheal
diverticulum mimicking an aggressive mediastinal lesion on FDG PET/CT: an interesting
case with review of the literature. Br J Radiol.
2012;85(1009):e17-21.http://dx.doi.org/10.1259/bjr/32814390
http://dx.doi.org/10.1259/bjr/32814390...
,
1111. Kokkonouzis I, Haramis D, Kornezos I, Moschouris H, Katsenos S,
Bouchara S. Tracheal diverticulum in an asymptomatic male: a case report. Cases J.
2008;1(1):181. http://dx.doi.org/10.1186/1757-1626-1-181
http://dx.doi.org/10.1186/1757-1626-1-18...
,
1414. Pinot D, Breen D, Peloni JM, Gaubert JY, Dutau H, Vervloet D. An
incidental finding in a 34-year-old male under investigation for haemoptysis.
Diagnosis: The radiological and endoscopic images demonstrate a complex defect along
the posterior tracheal wall consistent with acquired tracheal diverticulum. Eur
Respir J. 2009;33(5):1227-9. http://dx.doi.org/10.1183/09031936.00131908
http://dx.doi.org/10.1183/09031936.00131...
,
1717. Soto PC, Congregado M, Loscertales J. Divertículo traqueal adquirido
como causa de intubación orotracheal complicada. Arch Bronconeumol. 2012;48(2):64-6.
http://dx.doi.org/10.1016/j.arbres.2011.09.006
http://dx.doi.org/10.1016/j.arbres.2011....
)
Various approaches to the treatment of tracheal diverticulum have been described,
including surgical resection, endoscopic cauterization, and conservative measures. Most
tracheal diverticula are managed conservatively. Surgery is generally reserved for
larger diverticula or truly symptomatic presentations with recurrent
infections.(
11. Monroe MM, McLean M, Gross ND. Infected tracheal diverticulum
presenting as a cervical abscess Laryngoscope.
2013;123(7):1583-7.http://dx.doi.org/10.1002/lary.23978
http://dx.doi.org/10.1002/lary.23978...
,
33. Soto-Hurtado EJ, Pe-uela-Ruíz L, Rivera-Sánchez I, Torres-Jiménez J.
Tracheal diverticulum: a review of the literature. Lung.
2006;184(6):303-7.http://dx.doi.org/10.1007/s00408-006-0010-7
http://dx.doi.org/10.1007/s00408-006-001...
,
1313. Agustí EB, Vecina VM, Figuerola CR, Gaspà MM. Tracheal diverticulum:
a case report. Acta Otorrinolaringol Esp.
2007;58(6):278-9.http://dx.doi.org/10.1016/S2173-5735(07)70349-6
http://dx.doi.org/10.1016/S2173-5735(07)...
) Conservative management with antibiotics, mucolytics, and physiotherapy can
be appropriate for asymptomatic, elderly, or debilitated patients.(
22. Smelley CL, Bowen R, Nathan CO. Intermittently symptomatic tracheal
diverticulum: a case of a rare clinical phenomenon. Ear Nose Throat J.
2011;90(9):E10-2.
,
33. Soto-Hurtado EJ, Pe-uela-Ruíz L, Rivera-Sánchez I, Torres-Jiménez J.
Tracheal diverticulum: a review of the literature. Lung.
2006;184(6):303-7.http://dx.doi.org/10.1007/s00408-006-0010-7
http://dx.doi.org/10.1007/s00408-006-001...
,
1111. Kokkonouzis I, Haramis D, Kornezos I, Moschouris H, Katsenos S,
Bouchara S. Tracheal diverticulum in an asymptomatic male: a case report. Cases J.
2008;1(1):181. http://dx.doi.org/10.1186/1757-1626-1-181
http://dx.doi.org/10.1186/1757-1626-1-18...
,
1414. Pinot D, Breen D, Peloni JM, Gaubert JY, Dutau H, Vervloet D. An
incidental finding in a 34-year-old male under investigation for haemoptysis.
Diagnosis: The radiological and endoscopic images demonstrate a complex defect along
the posterior tracheal wall consistent with acquired tracheal diverticulum. Eur
Respir J. 2009;33(5):1227-9. http://dx.doi.org/10.1183/09031936.00131908
http://dx.doi.org/10.1183/09031936.00131...
) Here, we describe the case of a patient presenting with symptoms attributed
to a tracheal diverticulum.
Case report
A 69-year-old woman presented to the emergency room with a one-week history of fever, fatigue, and pleuritic chest pain. After clinical examination, she was discharged on a one-week course of amoxicillin-clavulanate and azithromycin. Despite the antibiotic therapy, she showed no clinical improvement over the following days, and, rather than getting better, she noticed a painless right neck mass complicated by dysphagia. In addition, detailed history-taking revealed a two-month history of hoarseness with no other associated complaint. Consequently, she was admitted to the hospital.
Her medical history was significant: pulmonary emphysema secondary to alpha-1 antitrypsin deficiency (PiZZ phenotype); bronchiectasis; recurrent pneumonia (at least two episodes per year); a thyroid neoplasm treated surgically (by thyroidectomy) in 2001; hypertension; and a herpes zoster infection. She reported no alcohol abuse or history of smoking. Her usual medications included levothyroxine, valsartan, hydrochlorothiazide, and bisoprolol.
On physical examination, she was afebrile with apparently normal skin. Chest auscultation revealed muffled heart sounds with an apical systolic murmur and diminished breath sounds. The remainder of the examination was unremarkable.
Laboratory tests revealed elevated inflammatory markers (leukocytes at 18,300/µL, with neutrophilia, and C-reactive protein at 31.01 mg/dL) and mild hyponatremia (132 mmol/L). A chest X-ray (at admission) showed a hypertransparent area in the upper mediastinum (Figure 1), and a CT scan of the chest (performed 10 days after admission) showed an air-filled multilocular mass measuring 3.5 × 2.5 cm, with no evidence of infection, in the right wall of the trachea, near the apex of the lung (Figure 2). The CT scan also confirmed the pulmonary insufflation, panacinar emphysematous lesions, and bronchiectasis described in previous examinations. Other than hyperemia, fiberoptic bronchoscopy revealed no abnormalities, showing no unequivocal communication with the tracheal lumen. Nevertheless, a diagnosis of tracheal diverticulum was proposed. A cervical magnetic resonance imaging (MRI) scan confirmed the cystic image and the resolution of the infection (Figure 3).
A CT scan of the chest at 10 days after admission, showing an air-filled multilocular mass (3.5 × 2.5 cm), with no evidence of infection, in the right wall of the trachea, near the apex of the lung.
Cervical magnetic resonance imaging at 10 days after admission, showing the cystic image and the resolution of the infection.
After a 14-day course of meropenem and vancomycin, the patient showed clinical improvement. Her inflammatory markers returned to normal values, and she was discharged from the hospital.
Discussion
The scarcity of reports on tracheal diverticulum is due partly to its rarity and partly
to the nonspecific nature of its symptoms, which tends to hinder the
diagnosis.(
22. Smelley CL, Bowen R, Nathan CO. Intermittently symptomatic tracheal
diverticulum: a case of a rare clinical phenomenon. Ear Nose Throat J.
2011;90(9):E10-2.
3. Soto-Hurtado EJ, Pe-uela-Ruíz L, Rivera-Sánchez I, Torres-Jiménez J.
Tracheal diverticulum: a review of the literature. Lung.
2006;184(6):303-7.http://dx.doi.org/10.1007/s00408-006-0010-7
http://dx.doi.org/10.1007/s00408-006-001...
4. Waite S, Sharma A, Machnicki S. Right paratracheal air cyst/tracheal
diverticulum.Appl Radiol. 2003;32(8):
-
55. Shah M, Joshi JM. Tracheal Diverticulum. Indian J Chest Dis Allied
Sci. 2012;54(1):39-40.
,
88. Sharma BG. Tracheal diverticulum: a report of 4 cases. Ear Nose
Throat J. 2009;88(1):E11.
,
99. Ortega HA, Vega NA, Comparini J, Carmo LV, Pereira, LP. Doença
cística traqueobrônquica. J Pneumol.
2001;27(6):349-54.http://dx.doi.org/10.1590/S0102-35862001000600010
http://dx.doi.org/10.1590/S0102-35862001...
,
1111. Kokkonouzis I, Haramis D, Kornezos I, Moschouris H, Katsenos S,
Bouchara S. Tracheal diverticulum in an asymptomatic male: a case report. Cases J.
2008;1(1):181. http://dx.doi.org/10.1186/1757-1626-1-181
http://dx.doi.org/10.1186/1757-1626-1-18...
,
1414. Pinot D, Breen D, Peloni JM, Gaubert JY, Dutau H, Vervloet D. An
incidental finding in a 34-year-old male under investigation for haemoptysis.
Diagnosis: The radiological and endoscopic images demonstrate a complex defect along
the posterior tracheal wall consistent with acquired tracheal diverticulum. Eur
Respir J. 2009;33(5):1227-9. http://dx.doi.org/10.1183/09031936.00131908
http://dx.doi.org/10.1183/09031936.00131...
) Tracheal diverticula are usually reported after the development of
complications or in postmortem examinations. (
11. Monroe MM, McLean M, Gross ND. Infected tracheal diverticulum
presenting as a cervical abscess Laryngoscope.
2013;123(7):1583-7.http://dx.doi.org/10.1002/lary.23978
http://dx.doi.org/10.1002/lary.23978...
,
44. Waite S, Sharma A, Machnicki S. Right paratracheal air cyst/tracheal
diverticulum.Appl Radiol. 2003;32(8):
,6 9,
1111. Kokkonouzis I, Haramis D, Kornezos I, Moschouris H, Katsenos S,
Bouchara S. Tracheal diverticulum in an asymptomatic male: a case report. Cases J.
2008;1(1):181. http://dx.doi.org/10.1186/1757-1626-1-181
http://dx.doi.org/10.1186/1757-1626-1-18...
,
1313. Agustí EB, Vecina VM, Figuerola CR, Gaspà MM. Tracheal diverticulum:
a case report. Acta Otorrinolaringol Esp.
2007;58(6):278-9.http://dx.doi.org/10.1016/S2173-5735(07)70349-6
http://dx.doi.org/10.1016/S2173-5735(07)...
,
1414. Pinot D, Breen D, Peloni JM, Gaubert JY, Dutau H, Vervloet D. An
incidental finding in a 34-year-old male under investigation for haemoptysis.
Diagnosis: The radiological and endoscopic images demonstrate a complex defect along
the posterior tracheal wall consistent with acquired tracheal diverticulum. Eur
Respir J. 2009;33(5):1227-9. http://dx.doi.org/10.1183/09031936.00131908
http://dx.doi.org/10.1183/09031936.00131...
) The largest case series, reported by Goo et al.,(
1010. Charest M, Sirois C, Carter Y, Rousseau J. Infected tracheal
diverticulum mimicking an aggressive mediastinal lesion on FDG PET/CT: an interesting
case with review of the literature. Br J Radiol.
2012;85(1009):e17-21.http://dx.doi.org/10.1259/bjr/32814390
http://dx.doi.org/10.1259/bjr/32814390...
) consisted of 64 confirmed cases.
Here, we have reported a case of a tracheal diverticulum associated with respiratory
tract infection, a right cervical mass, dysphagia and a two-month history of hoarseness.
The patient had a history of recurrent respiratory infections and chronic pulmonary
disease, specifically emphysema, which is known to be associated with tracheal
diverticulum. However, to our knowledge, this is the first report of alpha-1 antitrypsin
deficiency in a patient with tracheal diverticulum. It seems obvious that this was a
case of acquired tracheal diverticulum, given the presence of emphysema and the history
of thyroidectomy, which could have created weak points in the cervical anatomy,
increasing the propensity to develop diverticula. In addition, although we did not
perform a histological examination to confirm this hypothesis, the location of the
tracheal diverticulum was that most often reported for the acquired form.(
11. Monroe MM, McLean M, Gross ND. Infected tracheal diverticulum
presenting as a cervical abscess Laryngoscope.
2013;123(7):1583-7.http://dx.doi.org/10.1002/lary.23978
http://dx.doi.org/10.1002/lary.23978...
,
22. Smelley CL, Bowen R, Nathan CO. Intermittently symptomatic tracheal
diverticulum: a case of a rare clinical phenomenon. Ear Nose Throat J.
2011;90(9):E10-2.
,
88. Sharma BG. Tracheal diverticulum: a report of 4 cases. Ear Nose
Throat J. 2009;88(1):E11.
)
The treatment approach to tracheal diverticulum varies according to the age of the
patient, the clinical presentation, and the presence of comorbidities. Surgical
resection is typically reserved for younger, highly symptomatic patients, whereas older
patients, especially those with comorbidities, are treated conservatively.(
1212. Danielson GP, Jedlovszky V, Landigran GP. Tracheal diverticulum: a
rare finding in a patient with worsening chronic cough. Ear Nose Throat J.
2008;87(8):474-5.
) In the case presented here, the patient received conservative treatment.
Her history of recurrent infection (and consequent antibiotic courses) was probably
responsible for the lack of response to the initial treatment and the subsequent
requirement to switch to broad-spectrum antibiotics. Although the definitive diagnosis
was infected tracheal diverticulum, the CT and MRI, while still showing the air cyst,
indicated resolution of the infection, because they were both performed after at least
10 days of treatment with antibiotics. The fiberoptic bronchoscopy missed the point of
communication with the trachea and was not very helpful in the diagnosis, as has been
previously reported. (
33. Soto-Hurtado EJ, Pe-uela-Ruíz L, Rivera-Sánchez I, Torres-Jiménez J.
Tracheal diverticulum: a review of the literature. Lung.
2006;184(6):303-7.http://dx.doi.org/10.1007/s00408-006-0010-7
http://dx.doi.org/10.1007/s00408-006-001...
,
44. Waite S, Sharma A, Machnicki S. Right paratracheal air cyst/tracheal
diverticulum.Appl Radiol. 2003;32(8):
,
66. Han S, Dikmen E, Aydin S, Yapakci O. Tracheal diverticulum: a rare
case of dysphagia. Eur J Cardiothorac Surg.
2008;34(4):916-7http://dx.doi.org/10.1016/j.ejcts.2008.06.022
http://dx.doi.org/10.1016/j.ejcts.2008.0...
,
1010. Charest M, Sirois C, Carter Y, Rousseau J. Infected tracheal
diverticulum mimicking an aggressive mediastinal lesion on FDG PET/CT: an interesting
case with review of the literature. Br J Radiol.
2012;85(1009):e17-21.http://dx.doi.org/10.1259/bjr/32814390
http://dx.doi.org/10.1259/bjr/32814390...
,
1111. Kokkonouzis I, Haramis D, Kornezos I, Moschouris H, Katsenos S,
Bouchara S. Tracheal diverticulum in an asymptomatic male: a case report. Cases J.
2008;1(1):181. http://dx.doi.org/10.1186/1757-1626-1-181
http://dx.doi.org/10.1186/1757-1626-1-18...
,
1414. Pinot D, Breen D, Peloni JM, Gaubert JY, Dutau H, Vervloet D. An
incidental finding in a 34-year-old male under investigation for haemoptysis.
Diagnosis: The radiological and endoscopic images demonstrate a complex defect along
the posterior tracheal wall consistent with acquired tracheal diverticulum. Eur
Respir J. 2009;33(5):1227-9. http://dx.doi.org/10.1183/09031936.00131908
http://dx.doi.org/10.1183/09031936.00131...
,
1717. Soto PC, Congregado M, Loscertales J. Divertículo traqueal adquirido
como causa de intubación orotracheal complicada. Arch Bronconeumol. 2012;48(2):64-6.
http://dx.doi.org/10.1016/j.arbres.2011.09.006
http://dx.doi.org/10.1016/j.arbres.2011....
)
In conclusion, tracheal diverticula are mostly asymptomatic and frequently go undiagnosed. When symptomatic, the approach to treatment should be tailored to the patient. In many cases, as in the one reported here, conservative treatment is indicated, although a surgical approach can be considered in patients who do not respond to clinical treatment.
References
-
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» http://dx.doi.org/10.1002/lary.23978 -
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-
3Soto-Hurtado EJ, Pe-uela-Ruíz L, Rivera-Sánchez I, Torres-Jiménez J. Tracheal diverticulum: a review of the literature. Lung. 2006;184(6):303-7.http://dx.doi.org/10.1007/s00408-006-0010-7
» http://dx.doi.org/10.1007/s00408-006-0010-7 -
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-
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-
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» http://dx.doi.org/10.1016/j.ejcts.2008.06.022 -
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» http://dx.doi.org/10.1590/S0102-35862001000600010 -
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» http://dx.doi.org/10.1259/bjr/32814390 -
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» http://dx.doi.org/10.1186/1757-1626-1-181 -
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» http://dx.doi.org/10.1016/S2173-5735(07)70349-6 -
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» http://dx.doi.org/10.1183/09031936.00131908 -
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19Teh BM, Hall C, Kleid S. Infected tracheocoele (acquired tracheal diverticulum): case report and literature review. J Laryngol Otol. 2011;125(5):540-5.http://dx.doi.org/10.1017/S0022215110003026
» http://dx.doi.org/10.1017/S0022215110003026 -
20Buterbaugh JE, Erly WK. Paratracheal air cysts: a common finding on routine CT Examinations of the cervical spine and neck that may mimic pneumomediastinum in patients with traumatic injuries. AJNR Am J Neuroradiol. 2008;29(6):1218-21. http://dx.doi.org/10.3174/ajnr.A1058
» http://dx.doi.org/10.3174/ajnr.A1058
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Financial support: None
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*
Study carried out in the Department of Internal Medicine, Northern Lisbon Hospital Center, Hospital de Santa Maria, Lisbon, Portugal
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**
A versão completa em português deste artigo está disponível em www.jornaldepneumologia.com.br
Publication Dates
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Publication in this collection
Nov-Dec 2014
History
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Received
09 Apr 2014 -
Accepted
16 May 2014