Ann Thorac Surg 2008;85:e17-e19. doi:10.1016/j.athoracsur.2007.12.003
© 2008 The Society of Thoracic Surgeons
Case Reports
Two-Stage Tracheal Reconstruction of Primary Tracheal Non-Hodgkin Lymphoma With Nitinol Mesh Stent and Cervical Myocutaneous Flap
Chuang Cai, MD, PhDa,*,
Ren Chao Jiang, MDb,
Zhi Bin Li, MDb,
Xiao Dong Chen, MDb,
Mu Zhi He, MDb,
Lin Liu, MDb,
Xia Ying Zou, MDb
a Guangzhou Institute of Respiratory Disease, First Affiliated Hospital of Guangzhou Medical University, Guangzhou, China
b PLA Institute of Respiratory Disease, General Hospital of Guangzhou Military Command, Guangzhou, China
Accepted for publication December 3, 2007.
* Address correspondence to Dr Cai, Guangzhou Institute of Respiratory Disease, 151 Yanjiang Rd, Guangzhou, 510012, China (Email: rashcc{at}gmail.com).
 |
Abstract
|
|---|
Primary tracheal non-Hodgkin lymphoma is an extremely rare entity without consensus management strategy. We present a case of primary tracheal lymphoplasmacytoid lymphoma masquerading as asthma with wheezing and progressive dyspnea. A patented nitinol mesh stent was implanted in the right lateral cervical region 3 weeks before tumor resection. After 5.5-cm-long segmental tracheal resection, a neotracheal tube was constructed with cervical myocutaneous flap sandwiched around the implanted mesh stent, which was then anastomosed with the residual of the patient’s trachea. The patient has been well for 30 months.
|
Introduction
|
|---|
Primary tracheal non-Hodgkin lymphoma (PTNHL) is an extremely rare entity without definitive management strategy. In 2005 Takami and colleagues [1] reviewed only 28 cases of PTNHL from the published literature. We report a case of primary tracheal lymphoplasmacytoid lymphoma successfully treated by surgical resection and two-stage tracheoplasty.
A 42-year-old woman, who was a farmer, presented with a 1-year history of unproductive cough, wheezing, and exertional dyspnea. She had been diagnosed with asthma, but was unresponsive to inhaled corticosteroids and bronchodilators, as well as oral prednisone. She was referred for evaluation of progressive dyspnea on January 2005. She denied dysphagia, hoarseness, and hemoptysis. On admission, she had mild respiratory distress with respiration rate of 24 breaths/min. Her other vital signs were stable. Stridor was audible without auscultation. Lymphadenopathy and hepatosplenomegaly were negative.
Laboratory workup was unremarkable except for an increased serum immunoglobulin G level of 82.4 g/L. She had mild hypoxemia, with a PaO
2 of 88 mm Hg and fraction of inspired oxygen of 33%. Spirometry revealed extrathoracic airway obstruction with flattening of flow-volume loop, although result of laryngoscopy was negative.
The result of a chest roentgenogram was unremarkable, but a laminagram revealed a soft-tissue mass in the lower half of cervical trachea. Chest computed tomography (CT) scanning showed cervical-mediastinal tracheal stenosis without pulmonary and mediastinal involvement. An examination with a fibrobronchoscope demonstrated a circumferentially growing neoplasm 4 cm below the vocal cords and 3 cm above the carina (Fig 1) with approximately 70% occlusion of the tracheal lumen. Histopathology and histochemistry established low-grade lymphoplasmacytoid lymphoma. Results of CT scans of the abdomen and brain as well as gallium-citrate scintigraphy were unremarkable.
The patient was diagnosed with primary endotracheal B-cell NHL at stage IE by Ann Arbor staging. She underwent tracheostomy and biphasic tracheoplasty using a nitinol memory alloy mesh stent (Microport Medical, Shanghai, China).
Tracheal reconstruction was performed with slight modification of Zhao and colleagues’ protocol [2]. Briefly, a preshaped memory alloy mesh stent, with its bottom 3 to 5 cm from the right clavicle, was imbedded into a 4.5- x 8.0-cm subcutaneous pouch in the right lateral cervical region (Fig 2). Three weeks later, under general anesthesia, the affected portion of the trachea (11 cartilaginous rings, approximately 5.5 cm long, malignancy-free at both ends on frozen section) was excised with a reversed L-shaped incision, with care taken to protect the recurrent laryngeal nerve.
The mesh was covered with skin and subcutaneous tissues. The sternocleidomastoideus muscles were dissected at the middle point and freed toward the central one-third of the mesh area, keeping the lower one-third free. The artificial tracheal canal was constructed by rolling the sandwiched flap into a tube with vascularized cervical skin for the inner surface, mesh stent in the middle as the frame, and pedicled sternocleidomastoideus muscles as the outer layer. The flap was sutured with stainless steel wires at both ends and kept airtight by a continuous suture of pedicled sternocleidomastoideus muscles and subcutaneous layer (Figs 3a, b, c).
The neotracheal tube was turned upside down and imbedded on the original tracheal bed with fine steel threads, and the ends of the native trachea were sutured with the neotracheal tube by absorbable Vicryl (Ethicon, Somerville, NJ) suture (Figs 3c, d). The defect in the cervical region was mended with flap from the right anterior chest wall.
The patient recovered uneventfully, with successful tracheal recanalization revealed by fibrobronchoscopy 2 weeks later (Fig 4). At the 30-month follow-up, she had resumed menial labor without postoperative tracheal stenosis, infection, or local recurrence.
|
Comment
|
|---|
Primary tracheal malignancy is an uncommon entity, accounting for 0.1% of all malignancies. Primary tracheal NHL is even rarer, comprising 0.48% of tracheal tumors [1, 3, 4]. To our best knowledge, this is the second case of primary tracheal lymphoplamacytoid lymphoma [5]. Similar to other tracheal tumors, PTNHLs lack specific symptoms and often present with progressive dyspnea, stridor, and wheezing from central airway obstruction when the neoplasm causes more than 50% obstruction of tracheal lumen. They are prone to misdiagnoses of asthma or chronic obstructive pulmonary disease and are usually unresponsive to medical treatment with bronchodilators and steroids [1, 4]. These typical clinical features are displayed in the present case, with a longer and more indolent clinical course than that of tracheal squamous carcinoma and adenoid cystic carcinoma because most PTNHLs are of low-grade malignancy. Notably, hemoptysis, another common symptom of primary tracheal malignancy, is absent because the tracheal mucosa in most PTNHLs is still intact [1, 3, 4].
Because conventional chest roentgenograms are rarely diagnostic, the progressive dyspnea often promotes bronchoscopy or chest CT scanning, which is the best way to demonstrate the site, degree, and extent of tracheal stenosis and possible involvement of adjacent structures. Biopsy of affected mucosa by bronchoscopy with a comprehensive panel of imaging studies and bone marrow aspiration could provide pathologic subtyping of PTNHL with or without systemic dissemination [1, 4]. Most PTNHLs are B-cell lymphomas and usually respond with complete resolution after surgical resection, radiotherapy, or chemotherapy, or a combination [1, 4]. In view of its IE staging, adjuvant chemotherapy or radiotherapy was not given.
Although combined chemotherapy and radiotherapy is advocated for management of localized NHL, for early stage NHLs in the intestine, liver, colon, testis, and extradural region, surgery resection alone or with adjuvant chemotherapy is appropriate [6]. Because of its extreme rarity, management of PTNHL is still anecdotal.
For a primary tracheal malignancy, radical resection with curative intent is generally recommended when possible, with reliable eradication of malignancy [2]. But because most B-cell lymphomas are highly responsive to irradiation and chemotherapy, some clinicians favor tracheal-conservatory strategy, yet surgical resection is still used, especially to relieve life-threatening airway stenosis as in this case, allowing for the relatively slow-onset of chemotherapy or irradiation and possible reactive airway inflammation associated with irradiation [7]. Another issue is lymphoma recurrence with nonsurgical approaches, as demonstrated in a case in which surgical resection had to be used when PTNHL recurred after chemotherapy [4].
With these considerations and limitations, we decided to perform surgical resection and two-stage tracheoplasty, because primary end-to-end anastomosis entails high postoperative risks owing to the length of the lesion. The management of tracheal lesions with such length is still challenging despite advance in releasing maneuvers and experience with artificial tracheal tubes. Similar to the work of Zhao and colleagues [2] and Beldholm and colleagues [8], this approach with a nitinol memory alloy mesh stent as a frame and autografted cervical myocutaneous flap with sufficient blood supply (subthyroid artery) as the lining of the reconstructed tracheal tube enabled successful recanalization of the resected tracheal tube with maximal resumption of the normal function of the original trachea, such as epithelialization of the lining layer, satisfactory rigidity, and flexibility provided by the internal nitinol mesh stent with nice biocompatibility and plasticity, improved life quality, and less damage to cervical and hilar structures from avoidance of extensive releasing maneuvers for primary tracheostomy. Another advantage was minimal postoperative complications such as restenosis from granulation and infections from want of epithelialization that are often seen in other tracheal prosthesis.
Compared with the protocol of Beldholm and coworkers [8], using lower portion of the cervical skin, its subcutaneous layer, and pedicled sternocleidomastoideus muscles, our approach not only obviated the anastomosis of artery and veins but also ensured better reestablishment of local vascular and lymphatic circulation to the reepithelialized layer of the neotracheal tube. In addition to providing better blood supply, pedicled sternocleidomastoideus muscles rooted in the sternum and clavicle could help to fasten the neotracheal tube, especially in the early postoperative stage. It should also be noted that the appropriate distance (3 to 5 cm) from the bottom of the mesh stent to the right clavicle in the first stage will ensure better maneuverability of the myocutaneous flap and avoid excessive tension exerted on the imbedded neotracheal tube should the flap be too short.
Although much remains to be learned from this approach, it may open a newer field for the treatment of large defect of the trachea from malignancy or trauma.
|
Acknowledgments
|
|---|
We are indebted to Fan Rong for the illustrations about the procedure and Zeng Guang-qiao for technical assistance.
|
References
|
|---|
- Takami A, Okumura H, Maeda Y, et al. Primary tracheal lymphoma: case report and literature review Int J Hematol 2005;82:338-342.[Medline]
- Zhao F, Zhang Y, Liu S, Yu J. Artificial tracheal reconstruction with two-stage approach using memory-alloy mesh Chin Med J 2003;116:1949-1951.[Medline]
- Macchiarini P. Primary tracheal tumours Lancet Oncol 2006;7:83-91.[Medline]
- Fidias P, Wright C, Harris LH, et al. Primary tracheal non-Hodgkin’s lymphoma. A case report and review of the literature. Cancer 1996;77:2332-2338.[Medline]
- Gomez-Roman JJ, Perez-Montes R, Perez-Exposito MA, et al. Primary lymphoplasmacytoid lymphoma of the trachea with immunoglobulin G paraprotein Pathol Int 1999;49:1100-1104.[Medline]
- Coffey J, Hodgson DC, Gospodarowicz MK. Therapy of non-Hodgkin’s lymphoma Eur J Nucl Med Mol Imaging 2003;30:S28-S36.[Medline]
- Okubo K, Miyamoto N, Komaki C. Primary mucosa-associated lymphoid tissue (MALT) lymphoma of the trachea: a case of surgical resection and long term survival Thorax 2005;60:82-83.[Abstract/Free Full Text]
- Beldholm BR, Wilson MK, Gallagher RM, et al. Reconstruction of the trachea with a tubed radial forearm free flap J Thorac Cardiovasc Surg 2003;126:545-550.[Abstract/Free Full Text]
Top
Abstract
Introduction
