Tracheal diverticulum: A case report and literature review

Tracheal diverticulum: A case report and literature review

AM ER IC AN JOUR NA L OF OTOLARY NG OLOG Y –H EA D A N D N E CK ME D I CI NE AN D SUR G E RY XX ( 2 0 14 ) XXX –XXX Available online at www.sciencedi...

709KB Sizes 0 Downloads 68 Views

AM ER IC AN JOUR NA L OF OTOLARY NG OLOG Y –H EA D A N D N E CK ME D I CI NE AN D SUR G E RY XX ( 2 0 14 ) XXX –XXX

Available online at www.sciencedirect.com

ScienceDirect www.elsevier.com/locate/amjoto

Case report

Tracheal diverticulum: A case report and literature review☆ Haiping Lin, MM, Ziang Cao, MD⁎, Qing Ye, MD Department of Thoracic Surgery, Ren Ji Hospital, School of Medicine, Shanghai Jiao Tong University, Shanghai, China

ARTI CLE I NFO

A BS TRACT

Article history:

Purpose: Tracheal diverticulum (TD) is a rare, nonspecific symptom that is commonly

Received 17 December 2013

associated with other malformations in clinical presentation and appearance on imaging. The case presented and the literature review performed highlight the importance of combining 3 D reconstruction technology and computerized Tomography (CT) results to better characterize TD on the list of differential diagnoses of patients. Methods: The case described is of a 44-year-old male with a 1-year history of repeatedly coughing with white phlegm. Computerized tomography and three dimension reconstruction technology were employed to diagnose tracheal abnormalities. The patient underwent surgical resection of the tracheal diverticulum. Reexamination of the neck, chest, trachea and lungs of the patient was performed with CT scan post operation. Results: Chest CT confirmed the presence of the air cavity lesion behind the neck segment and may be the trachea cysts. Bronchoscope examination was all normal. Furthermore, HRCT scan and the tracheal reconstruction were performed (as shown in Fig. 1), which show cystic cavity lesion was on right rear trachea and a tiny tha was connected the tracheal posterior wall. Post-operation, reexamination showed that the neck, chest CT, trachea and lungs had no detectable abnormity. Conclusion: Diagnostic techniques such as HRCT and 3D reconstruction technology may help to diagnose the tracheal diverticulum timely and accurately. Resection of the diverticulum is the proper surgery, but only for symptomatic congenital diverticulum; therefore preoperative definite classification is important (acquired or congenital diverticulum). © 2014 Elsevier Inc. All rights reserved.

1.

Introduction

Tracheal diverticulum is a benign entity characterized by single or multiple invaginations of the tracheal wall. Tracheal diverticulum resembles laryngeal diverticulum, which is a more common disorder and may share the same pathogenic mechanism [1]. Tracheal diverticulum was first described by Rokitansky in 1838 [2], with few cases reported since. The largest series of 64 cases was reported by Goo et al. [3]. The

overall prevalence is about 1% according to an autopsy series by MacKinnon [4], who found 8 cases in 867 routine serial autopsies, and 0.3% in children over 10 years of age according to fiberoptic bronchoscope studies [5], although it is rarely reported in clinical practice. In addition to detecting the diverticulum, the diagnosis of this rare entity is made by CT scan of trachea (preferably Spiral/Helical CT) and re-construction in varying angles in coronal plane to visualize communication with tracheal wall.



Conflicts of interest: All authors declare that they have no conflicts of interest. ⁎ Corresponding author at: Department of thoracic surgery of Renji hospital, School of medicine, Shanghai Jiaotong University, No.1630 Dongfang Road, Shanghai, 200127, China. Tel./fax: +86 21 68383765. E-mail address: [email protected] (Z. Cao). http://dx.doi.org/10.1016/j.amjoto.2014.03.015 0196-0709/© 2014 Elsevier Inc. All rights reserved.

Please cite this article as: Lin H, et al, Tracheal diverticulum: A case report and literature review, Am J Otolaryngol–Head and Neck Med and Surg (2014), http://dx.doi.org/10.1016/j.amjoto.2014.03.015

2

AM ER IC AN JOURNAL OF OTOLARYNGO LOGY –H EAD AN D N E CK ME D I CI NE AN D SUR GE RY XX ( 2 0 14 ) XXX–X XX

cardiac auscultation difference. Chest computerized Tomography (CT) confirmed the presence of the air cavity lesion behind the neck segment and may be the trachea cysts. Bronchoscope examination was all normal. Furthermore, HRCT scan and the tracheal reconstruction were performed (as shown in Fig. 1), which showed cystic cavity lesion was on right rear trachea and a tiny tha was connected the trachea posterior wall. Hospital diagnosis: tracheal diverticulum.

3.

Fig. 1 – Helicoidal CT, 3-mm axial slice.Air image of ovoid morphology which is in intimate contact with the right tracheal wall.

Computerized tomography (CT) provides information concerning the location, origin, and size of the lesion, thus helping to distinguish between congenital and acquired lesions depending on the presence or absence of cartilage and the size of the neck of the diverticulum [6]. The development of new techniques such as three-dimensional reconstruction may be beneficial in the future for the morphologic diagnosis of this entity. There are also reports confirming the diagnosis of an unsuspected tracheal diverticulum at surgery or autopsy [7]. Treatment options include surgical resection, which can be performed via a lateral cervical approach without the need for thoracotomy, endoscopic cauterization with laser or electrocoagulation, and conservative management (antibiotics, mucolytic agents and physiotherapy). Excellent results have been reported after surgical excision. The option depends on the patient’s physical state, age, and symptoms, with conservative management being more recommended for elderly patients, reserving surgery for the younger cases [8,9].

2.

Course of treatment

Preoperative preparation and then performed the tracheal diverticulum resection. After general anesthesia in a supine position with head back, indwelling gastric tube, layer-by-layer cut cervical and root collar incision, exposed trachea, carefully detected the abnormality at the right side of the trachea, and then the laryngeal recurrent nerve on the right side was freed out with a rubber band pulling open. After careful, a pouch lesion was identified behind the tracheaesophageal groove of trachea, appears void status with tiny tha connected with trachea membrane. A complete resection of the pouch lesion was performed and sutured with absorbable thread through adjoining neighbor trachea membrane. In addition, there was a nodular lesion in the upper part of the diverticulum, the intraoperative frozen pathological examination after resection showed chronic lymphadenitis. After the wound was checked with no bleeding and the esophagus with no damage, the incision was closed step by step. The final pathological report: tracheal mucosa tissue and smooth muscle tissue appeared in the wall of the cyst. The final diagnosis: congenital tracheal diverticulum. The patient went out of hospital four days after operation. Six-month follow-up after surgery displayed no symptoms of cough and expectoration. Reexamination showed that the neck, chest CT, trachea and lungs had no detectable abnormity (as shown in Fig. 2).

Case report

The patient has given his informed consent prior to his inclusion in the study, and the studies have been approved by Renji hospital, School of medicine, Shanghai Jiaotong University Ethics Committee and performed in accordance with the ethical standards. The case described is of a 44-year-old male with a 1-year history of repeatedly coughing with white phlegm. There were no fever, no yellow purulent sputum and no blood in phlegm in the course. The patient previously took drugs such as antibiotics, mucosolvan, and licorice mixture, yet no symptom significantly improved. The patient was healthy and denied cough for a long time and smoking history. Physical examination: the neck without tumor or lymph node enlargement, breath sounds crude in double lung, no rale and no

Fig. 2 – Helicoidal CT, 3-mm axial slice at six-month follow-up after surgery.Reexamination showed that the neck, chest CT, trachea and lungs were normal.

Please cite this article as: Lin H, et al, Tracheal diverticulum: A case report and literature review, Am J Otolaryngol–Head and Neck Med and Surg (2014), http://dx.doi.org/10.1016/j.amjoto.2014.03.015

AM ER IC AN JOUR NA L OF OTOLARY NG OLOG Y –H EA D A N D N E CK ME D I CI NE AN D SUR G E RY XX ( 2 0 14 ) XXX –XXX

4.

Discussion

In recent years, cases of tracheal diverticulum have been reported to be gradually increasing by radiologists. The characteristics of tracheal diverticulum for CT thin layer scanning were gas or liquid lumen of the trachea was identified, usually 2 cm in diameter. In some cases, lumen was connected with the trachea via some small pipeline. With the above characteristics of imaging diagnosis, we can conclude it as tracheal diverticulum. But for how to treat this kind of disease, there still very rare publication reported by thoracic surgery doctors. Based on the etiology and pathology analysis, tracheal diverticulum can be divided into two types: congenital diverticulum and acquired diverticulum [10]. Congenital diverticulum arises from a defect in endodermal differentiation during development of the membranous posterior tracheal wall, or else from a defect in the development of the tracheal cartilage during the sixth week of fetal life [11]. The Congenital diverticulum possesses a complete tracheal anatomy (respiratory epithelium, smooth muscle, and cartilage) and is usually filled with mucus. It occasionally accompanies other congenital malformations such as trachea-esophageal fistula [12]. Acquired tracheal diverticula may appear at any level, though they are more common in the poster lateral region [5,13,14]. Acquired tracheal diverticula are larger than congenital tracheal diverticula and have a wider opening. Acquired tracheal diverticula may be single or multiple. They originate as the result of increased intraluminal pressure that results in herniation of the mucous membrane through a weak point in the tracheal wall, accompanied by chronic cough, e.g., chronic obstructive pulmonary disease [2,15]. The wall in acquired tracheal diverticula, unlike in congenital diverticula, is formed solely of respiratory epithelia and lacks smooth muscle and cartilage. The way of surgical treatment of tracheal diverticulum is rarely reported by literature. Our experience is that when a lesion nearby the lumen is identified by CT examination, CT manifestations should be carefully observed, and the history of disease should be inquired. According to the symptoms and the imaging characteristics of diverticulum, we carried out the preliminary classification. Multiple and wide base diverticula, especially for patients with chronic cough symptoms such as chronic bronchitis, should be considered as acquired tracheal diverticula. The removal of the diverticulum is not the best choice for the treatment and the surgical resection has no obvious benefit. For the protopathy, prevention of the infection of diverticulum is the optimum selection. For congenital diverticulum, due to the long-term accumulated mucous in the lesion, which could be the source of infection, and diverticulum resection is needed. Based on the characteristics of the symptoms of the patient and the phenotype of CT image in our study, it can be diagnosed as congenital tracheal diverticulum; the diverticulum wall can be normal tracheal tissue and secrete mucus also. When mucus in the diverticulum goes through the airway of lumen, it can lead to cough and phlegm. The symptom of the patient was significantly improved post

3

operation which also confirmed that our preoperative assessment is correct. The diverticulum was small and usually is closed; therefore the preoperative bronchoscope examination was normal. For the surgical path, in congenital diverticulum, it is located in the border of cervical and thoracic trachea. Before surgery, the patient lay his head back with CT scan examination, and the position of diverticulum was above the entrance of the thorax which was suitable for the neck incision surgery. Although preoperative CT implies that diverticulum is appeared a gas filling state, in the same time, patients without spontaneous breathing, positive pressure disappeared in the upper diverticulum, repeated probe action extrusion and an empty state was formed for internal gas is squeezed into tracheal cavity. In addition, it brought difficulties for intraoperative dissection, and needed to avoid damage of the laryngeal recurrent nerve and the crucial tissue around esophagus. Our approach was indwelling gastric tube pre-operation which can indicate clearly the position of esophagus during intraoperative period for avoiding injuring the wall of esophagus. If the tracheal diverticulum is difficult to observe, we made it become visible in trachea-esophageal groove and isolated laryngeal recurrent nerve, which was better for protection based on the isolation of diverticulum behind a trachea.

5.

Conclusions

In summary, tracheal diverticulum is a rare clinical disease. Diagnostic techniques such as HRCT and 3D reconstruction technology may help to diagnose the tracheal diverticulum timely and accurately. However, diverticulum in the upper esophageal (Zenker diverticulum) was totally different with tracheal diverticulum. Resection of the diverticulum is the proper surgery, but only for symptomatic congenital diverticulum; therefore preoperative definite classification is important (acquired or congenital diverticulum). In addition, we should pay attention to avoid damage of the laryngeal recurrent nerve and the esophageal.

REFERENCES

[1] Infante M, Mattavelli F, Valente M, et al. Tracheal diverticulum: a rare cause and consequence of chronic cough. Eur J Surg 1994;160:315–6. [2] Frenkiel S, Assimes I, Rosales J. Congenital tracheal diverticulum. A case report. Ann Otol Rhinol Laryngol 1979;89:406–8. [3] Goo JM, Im JG, Ahn JM, et al. Right paratracheal air cysts in the thoracic inlet: clinical and radiologic significance. AJR Am J Roentgenol 1999;173:65–70. [4] MacKinnon D. Tracheal diverticula. J Pathol Bacteriol 1953;65:513–7. [5] Caversaccio M-D, Becker M, Zbären P. Tracheal diverticulum presenting with recurrent laryngeal nerve paralysis. Ann Otol Rhinol Laryngol 1998;107:362. [6] Kwong JS, Muller NL, Miller RR. Diseases of the trachea and main-stem bronchi: correlation of CT with pathologic findings. Radiographics 1992;12:645–57. [7] Henderson CG, Harrington RL, Izenberg S, et al. Tracheocele after routine tracheostomy. Otolaryngol Head Neck Surg 1995;113:489–90.

Please cite this article as: Lin H, et al, Tracheal diverticulum: A case report and literature review, Am J Otolaryngol–Head and Neck Med and Surg (2014), http://dx.doi.org/10.1016/j.amjoto.2014.03.015

4

AM ER IC AN JOURNAL OF OTOLARYNGO LOGY –H EAD AN D N E CK ME D I CI NE AN D SUR GE RY XX ( 2 0 14 ) XXX–X XX

[8] Dutta C, Basmajian J. Tracheal diverticula. Report of a specimen and review of the literature. Surgery 1960;48:335. [9] Mathur N, Sardana P, Singh V, et al. Adult tracheocele with large cervical presentation. J Laryngol Otol 1999;113:364–5. [10] Soto-Hurtado EJ, Peñuela-Ruíz L, Rivera-Sánchez I, et al. Tracheal diverticulum: a review of the literature. Lung 2006;184:303–7. [11] Tanaka H, Mori Y, Kurokawa K, et al. Paratracheal air cysts communicating with the trachea: CT findings. J Thorac Imaging 1997;12:38–40.

[12] Bhatnagar V, Lal R, Agarwala S, et al. Endoscopic treatment of tracheal diverticulum after primary repair of esophageal atresia and tracheoesophageal fistula. J Pediatr Surg 1998;33:1323–4. [13] Pérez JMH, Negrín LP, Martín JJB, et al. Intratracheal diverticulum. J Bronchology Interv Pulmonol 2005;12:104–5. [14] Restrepo S, Villamil MA, Rojas IC, et al. Association of two respiratory congenital anomalies: tracheal diverticulum and cystic adenomatoid malformation of the lung. Pediatr Radiol 2004;34:263–6. [15] Rahalkar M, Lakhkar D, Joshi S, et al. Tracheal diverticula—report of 2 cases. Indian J Radiol Imaging 2004;14:197.

Please cite this article as: Lin H, et al, Tracheal diverticulum: A case report and literature review, Am J Otolaryngol–Head and Neck Med and Surg (2014), http://dx.doi.org/10.1016/j.amjoto.2014.03.015