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figure 15-5 Flow volume loops in a 57-year-old man with a 7-year history of progressive wheezing, dyspnea, cough, and respiratory infections. Preoperative on left; 2 months postoperative on right. Values for pulmonary function tests are in Table 15-1. The patient's bronchoscopic findings are displayed in Figures 15-4C,D,E.

figure 15-5 Flow volume loops in a 57-year-old man with a 7-year history of progressive wheezing, dyspnea, cough, and respiratory infections. Preoperative on left; 2 months postoperative on right. Values for pulmonary function tests are in Table 15-1. The patient's bronchoscopic findings are displayed in Figures 15-4C,D,E.

The goiter may be so large and of such long duration that unilateral vocal cord paralysis has resulted from pressure defunctioning of a recurrent laryngeal nerve. Nerve paralysis or malfunction may also be the result of prior thyroid surgery, which many of these patients have undergone. Thyroid function should always be measured, since a few patients may indeed be hyperthyroid. Isotopic thyroid scans are not of particular value since the goiters may well be nonfunctional. Pulmonary function tests can provide information about fixed upper airway obstruction and pulmonary parenchymal disease. However, even patients with very limited pulmonary function and other severe medical problems can usually safely undergo the requisite operation, with dramatic functional improvement. Cervical and thoracic CT scans demonstrate the distribution and extent of the intrathoracic goiter and are always advisable (Figure 15-7).

figure 15-6 Intrathoracic (substernal) goiter in a 72-year-old man with a history of dyspnea, dysphagia, and stridor for several years. Chest roentgenograms. A, Posteroanterior view showing marked displacement of the larynx and trachea to the right, with marked tracheal compression. B, Oblique view emphasizes severe tracheal deviation and compression, which reduced the airway cross section by over 80%. Complete relief followed removal of the bilateral adenomatous goiter by right total and left subtotal thyroidectomy via a collar incision and upper partial sternotomy.

figure 15-6 Intrathoracic (substernal) goiter in a 72-year-old man with a history of dyspnea, dysphagia, and stridor for several years. Chest roentgenograms. A, Posteroanterior view showing marked displacement of the larynx and trachea to the right, with marked tracheal compression. B, Oblique view emphasizes severe tracheal deviation and compression, which reduced the airway cross section by over 80%. Complete relief followed removal of the bilateral adenomatous goiter by right total and left subtotal thyroidectomy via a collar incision and upper partial sternotomy.

No matter how tortuous the airway appears on imaging studies, it has always been possible to pass a rigid bronchoscope through the trachea, straightening the airway out as the bronchoscope is advanced (Figure 15-8). An endotracheal tube can always be passed into the trachea, despite its tortuosity, and if need be, over a flexible bronchoscope or over a tube changer placed via a rigid bronchoscope. The difficulty that may be encountered is that of initiating bronchoscopy or laryngoscopy because of marked displacement and torsion of the larynx by the goiter. If difficulty in intubation is anticipated because of the size of the goiter, it is wise to have a rigid bronchoscope available. This can always be passed, even where it is difficult to identify a markedly distorted and deviated larynx with a flexible instrument. In an extremely difficult case, a laryngoscope is useful to help introduce the rigid bronchoscope into the glottis. If the trachea is markedly compressed, intubation is preferable to the compromise of laryngeal mask airway.

Operation is accomplished through a low collar incision, which may have to be longer than usual, depending upon the size of the goiter. In a number of patients, upper sternotomy becomes necessary in order to extract the intrathoracic component without undue bleeding. Only rarely, principally in patients who have had previous goiter surgery that extended intrathoracically, is a complete median sternotomy needed. In 2 unique cases of a very extensive goiter complicated by previous operations, further extension of the incision into the right hemithorax through the fourth interspace was also required. In huge goiters with posterior and inferior extensions, it is prudent to prepare and drape a wide operative field for possible incisional extension.

In most patients, removal of the goiter is all that is required to relieve tracheal and esophageal compression. On a rare occasion, cartilage thinning is so severe that the airway collapses and the patient becomes even more dyspneic following thyroidectomy. Usually, the trachea opens up when the mass is removed and the cartilages gradually return to a more normal shape and stabilize in the postoperative weeks. However, if

figure 15-8 Bronchoscopy showing marked bilateral tracheal compression by a goiter. Although the tracheal rings are difficult to see in the region of maximal deformation, enough substance remained to provide an airway of over 50%, immediately after thyroidectomy, without collapse on respiration. Also see Figure 41 (Color Plate 16).

a now severely malacic trachea has lost the rigid splinting of the surrounding goiter, it can collapse more completely. The progression is from mass obstruction to that of malacic collapse. In order to recognize this possibility, the endotracheal tube is withdrawn intraoperatively, after thyroidectomy, to a point just below the glottis. The trachea is examined and palpated in the open operative field and the trachea is observed during respiration through a flexible bronchoscope passed through the endotracheal tube. It is helpful if the patient makes spontaneous respiratory efforts during examination. Usually, the airway appears adequate, although somewhat deformed, and no further reinforcing procedures are needed.

If this assessment proves incorrect and the patient becomes dyspneic following extubation at the close of operation, then the patient should promptly be relaryngoscoped and bronchoscoped. The possibility of recurrent laryngeal nerve damage causing glottic inadequacy must first be ruled out. The trachea is then examined carefully for evidence of malacic collapse and obstruction. For either problem, my preferred management is to pass an uncuffed endotracheal tube of smaller diameter to maintain a satisfactory postoperative airway. In a few days, after tissue planes are well coapted, the endotracheal tube may be removed over a flexible bronchoscope, preferably in the operating room, and the trachea and larynx reassessed. If obstruction persists, a tracheostomy may now be performed safely through the sealed tissue planes. It should be noted, however, that Lahey and Hoover did not observe infection in their patients after tracheostomy was done for tracheal collapse at the initial thyroid operation.11 In the rare event of malacia, a T tube is preferable to a tracheostomy tube for both function and comfort. Over a period of weeks and months, the trachea usually becomes firm again, permitting removal of the T tube. Since the cartilages usually do regain stability, permanent expandable stents are inadvisable. Coated expandable stents are prone to producing rings of granulations at either end. As the incidence of massive goiter has diminished, critical tracheal collapse after a thyroidectomy is rarely encountered. In earlier days, failure of prompt recognition and treatment could even result in fatalities.12

If severe malacia is identified during surgery, then it may be managed safely, as just described. An alternative is to place splinting polypropylene rings in channels around the malacic segment of trachea, embedding them with strap muscles (see Chapter 32, "Surgery for Tracheomalacia, Tracheopathia Osteoplastica, Tracheal Compression, and Staged Reconstruction of the Trachea"). I have done this in a few patients without long-term difficulty. Circumferential wrapping of the trachea with a heavy Marlex mesh attached with tissue adhesive has been reported, but I believe one must be concerned about possible devascularization.13 Another procedure that was used in the past to solve this problem, in then areas of endemic goiter in Europe, was placement of traction sutures through the wall of the trachea, which were tied over buttons resting either on the surface of the sternocleidomastoid muscles on either side or on the skin. Externally tied traction sutures were removed after the trachea appeared to be stable. I have had no experience with such technique.

Special care must be taken to spare the recurrent laryngeal nerves and the parathyroid glands, since the bulk of large goiters often thwart their easy identification.

Cysts

Cysts that can compress the trachea are bronchogenic, thymic, and parathyroid.14-19 Bronchogenic cysts are more likely to produce tracheobronchial symptoms in children, perhaps because the juvenile trachea is more malleable. Life-threatening distress can occur in infants.20 Foregut enteric cysts are rare and may present as air-filled lesions adjacent to the trachea in its upper portion or at the thoracic inlet. If large enough, such cysts cause cough or an uncomfortable sensation of a mass in the neck. Esophageal compression may produce dysphagia. A cyst may be adherent to the tracheal wall, but communication to the trachea may be tiny or absent (Figure 15-9). Such cysts are presumed to be of congenital origin, but histology of the lining cells may not clearly differentiate between a tracheal or esophageal origin. The cyst must not be confused with herniation of the lung apex through Sibson's fascia or with apical bullae.

figure 15-9 Tracheal cyst. Dorsal enteric cyst, lined by respiratory-type epithelium, in a 61-year-old woman with dysphagia accompanied by gurgling, occasional dysphonia, and sense of a mass. She had no respiratory symptoms. The cyst was attached firmly to the tracheal wall at the right posterior cartilaginous margin, and lay at the level of the thoracic inlet. It was air filled, but communication with the trachea was too small to identify. The right recurrent laryngeal nerve was tightly adherent to the surface of the cyst. A, Exterior of cyst. B, Cyst on section.

figure 15-9 Tracheal cyst. Dorsal enteric cyst, lined by respiratory-type epithelium, in a 61-year-old woman with dysphagia accompanied by gurgling, occasional dysphonia, and sense of a mass. She had no respiratory symptoms. The cyst was attached firmly to the tracheal wall at the right posterior cartilaginous margin, and lay at the level of the thoracic inlet. It was air filled, but communication with the trachea was too small to identify. The right recurrent laryngeal nerve was tightly adherent to the surface of the cyst. A, Exterior of cyst. B, Cyst on section.

Suen and colleagues found mild dyspnea in only 1 of 42 patients with bronchogenic cysts, 9 of which were carinal and 9 peritracheal.14 Respiratory distress occurred in 6 of 8 patients with cysts at the carina, with extrinsic airway compression found in all.15 St. Georges and colleagues, in a multi-institutional study of 86 bronchogenic cysts, noted dyspnea in 16 which had mediastinal localization and in 8 with pulmonary cysts; specific airway compression was not mentioned.21 Ribet and colleagues found dyspnea to be more common in 24 infants and children with bronchogenic cysts (25%) than in 45 adults (4 to 11%).22 Compression was radiologically seen against the trachea in 5, against the left main bronchus in another 5, the right main bronchus in 2, and the intermediate bronchus in 3 patients.

Thoracotomy, most often posterolateral and on the side where the cyst presents, remains the favored surgical approach to bronchogenic cysts.14,15,21,22 Median sternotomy is useful on occasion for a cyst deemed more accessible by this route, or was used in earlier cases prior to CT definition when a diagnosis of mediastinal tumor was entertained. Complete excision of a cyst prevents recurrence. Incomplete removal can result in infection, a chronic draining sinus, or even fistula to a bronchus or to esophagus. Secondary removal may prove technically very difficult (Figure 15-10).

Removal via cervicotomy or mediastinoscopy is applicable only in special circumstances. Communication from a cyst to a bronchus is usually managed by closure of a small opening, with second layer reinforcement. Bronchial sleeve resection and pericardial patch repair have been employed for larger defects.15,23 In one 8/2- year-old girl, a bronchogenic cyst in the carinal location, which had been excised elsewhere, proved to contain adenocarcinoma. Carinal resection and reconstruction was therefore subsequently performed, with apparent long-term cure.14

Thymic cysts are rare, but can occur in the neck and upper mediastinum, or, more often, entirely within the anterior mediastinum.18 Thyroid and parathyroid tissues have also been found in these cysts. A true

figure 15-10 Persistent bronchogenic cyst in subcarinal location in a 29-year-old male, 4 years after incomplete removal. Two years after the initial operation, following episodes of mediastinal infection and mediastinal drainage, a broncho-esophageal cystic fistula precipitated esophageal diversion. Intermittent signs of bronchial fistula, esophageal fistula, and sinus tract drainage led to referral. The sinogram shows a residual cyst with contrast entering the bronchus intermedius (arrow). The entire cyst was dissected out, the bronchial fistula closed, the esophagus repaired at the site of a densely scarred fistula, and the suture lines buttressed with healthy pericardial fat pad. Recovery was uneventful.

figure 15-10 Persistent bronchogenic cyst in subcarinal location in a 29-year-old male, 4 years after incomplete removal. Two years after the initial operation, following episodes of mediastinal infection and mediastinal drainage, a broncho-esophageal cystic fistula precipitated esophageal diversion. Intermittent signs of bronchial fistula, esophageal fistula, and sinus tract drainage led to referral. The sinogram shows a residual cyst with contrast entering the bronchus intermedius (arrow). The entire cyst was dissected out, the bronchial fistula closed, the esophagus repaired at the site of a densely scarred fistula, and the suture lines buttressed with healthy pericardial fat pad. Recovery was uneventful.

thymic cyst is to be differentiated from a cystic thymoma, although a small thymoma or even carcinoma has been found in the cyst wall. In a review of 46 patients, Graeber and colleagues did not find tracheal compression, but rather dysphagia, hoarseness, and pain as manifestations.17 This is probably due to the generally anterior location of cysts in front of the great vessels and pericardium. Cervical presentation in infancy or childhood appears more likely to cause tracheal obstruction.16 In 1 child treated for severe tracheal compression by a thymic cyst, sufficient malacia proved to be present after cystectomy to require prolonged stenting with a tracheostomy tube until the cartilages became firmer (see Figure 6-16 in Chapter 6, "Congenital and Acquired Tracheal Lesions in Children"). Parathyroid cysts are, on occasion, sufficiently large to narrow the trachea when they are in a cervical location or present as a cervical extension from the anterior-superior mediastinum.18,19 A retrotracheal position is also described. The airway is not often affected symptomatically. In a few patients, hyperparathyroidism is present. The lesions are quite visible on roentgenograms and CT scans, and can easily be mistaken to be of thymic or thyroid origin. Surgical removal is likely to be effected through a cervical incision with, at most, an upper sternal extension.

Neoplasms

Airway invasion by secondary tumors (ie, carcinomas of the thyroid, lung, and esophagus) is discussed in Chapter 8, "Secondary Tracheal Neoplasms." Mediastinal tumors, including germ cell tumors, hemangiopericytomas, thymomas, and lymphomas, are infrequent causes of tracheal obstruction. In the case of critical tracheal compression by a massive mediastinal tumor, a biopsy by core needle aspiration may be safer than subjecting the patient even to a brief general anesthesia for a diagnostic open biopsy. Cardiodynamic collapse has, on occasion, occurred under general anesthesia with absolutely massive tumors infiltrating the mediastinum, encasing the aortic root, and great vessels, despite a fully patent airway guaranteed with an endotracheal tube. The origin may be reflexive.

Hyalin-Vascular Lymph Node Hyperplasia

(Angiofollicular Lymph Node Hyperplasia; Castleman's Disease)

A 69-year-old woman with a 4-month history of rapidly progressive dysphagia, dyspnea with stridor, and cervical swelling, presented with massive involvement of central mediastinal lymph nodes, shown by biopsy to be Castleman's disease. The lobulated disease narrowed and displaced the trachea to the right and posteriorly, impinging on the carina and narrowing main stem bronchi. The esophagus was displaced to the right. Aortography and venography showed brachiocephalic, left common carotid, and subclavian arteries displaced, and subclavian and brachiocephalic veins obstructed with thrombus. Extensive collateral venous flow in the neck and posterior mediastinum accompanied the superior vena cava syndrome.

The patient responded symptomatically to irradiation treatment (2,800 cGy), with improved breathing and swallowing, but died from bilateral pneumonia, likely associated with severe radiation pneumonitis, 2 months later.

Dr. Benjamin Castleman (personal communication) described 2 prior patients with bronchial compression from this disease, but without superior vena cava syndrome. One had responded to irradiation. Judging from other mediastinal masses that have been excised and proved to be Castleman's disease, surgical removal would have been the preferable treatment in the patients described above.

Tracheocele and Diverticula

Air-containing outpouchings adjacent to the trachea have been described by many terms, including tracheocele, tracheal diverticula, tracheal diverticulosis, trachiectasis, aerocele, and aerial goiter (Figure 15-11). These rare diverticula have been classified by Katz and colleagues as follows:24

1) Rudimentary bronchus. These rare and sometimes multiple diverticula involve all layers of the tracheal wall and occur at the junction of the membranous and cartilaginous wall, especially on the right. They are asymptomatic for the most part.

2) Ovoid diverticula, which appear pedunculated, are filled with viscous secretions and may be related to cystic dilation of mucus glands. They are asymptomatic.

3) A single air-filled sac, which usually has a wide mouth and may fill with enough air to produce symptoms, is seen rarely. It is of tracheal origin and differentiated from the slightly more common but still rare laryngocele. It may be associated with chronic cough.

4) Outpouchings of the thin tracheal wall, between deformed and partly malacic cartilaginous rings in tracheobronchomegaly (see Chapter 14, "Infectious, Inflammatory, Infiltrative, Idiopathic, and Miscellaneous Tracheal Lesions"), are seen as wide-mouthed diverticula on bronchoscopy and on imaging.

Symmetric Lipomatosis Colli

Symmetric lipomatosis colli (Lanois-Bensaude adenolipomatosis, multiple or benign symmetric lipomatosis, lipoma annulare colli, Madelung's disease), affecting the neck, supraclavicular regions, and upper back, very rarely is accompanied by respiratory distress due to laryngeal compression or infiltration of the false vocal cords and by tracheal compression from mediastinal fatty infiltration.25,26 The fat may sometimes enlarge rapidly, or it may remain unchanged for long periods. It is not encapsulated and penetrates surrounding structures deeply. Familial occurrence has been reported and mitochondrial DNA mutations have been implicated.27 Multiple metabolic abnormalities and peripheral autonomic neuropathy have been found in association with the disease.26

The occurrence of airway difficulty is exceedingly rare in an already rare condition. The disease is observed in adult life and sometimes seems to be associated with large alcohol intake, but for the most part, it leads principally to disfigurement, limitation of movement of the neck and upper extremities, and difficulty in fitting clothing.28 Other symmetric fat deposits occur in some patients in the breast, abdomen, lower back, and upper thighs.26 Enzi found mediastinal involvement in 5 of 19 patients, confirmed by CT scans.26 Four had clinical signs and symptoms of airway compression, and one who had severe narrowing of the trachea and superior vena cava obstruction as well, required a tracheostomy. A recurrent nerve was also paralyzed in this patient.

A collar of fatty tissue encircles the neck, lobular masses of fat fill the supraclavicular spaces, a "buffalo hump" is present in the back of the neck, and "football suit" shoulders are seen, summarized descriptively as a "horse collar" distribution (Figure 15-12). Parotid area deposits may produce a "chipmunk" appearance. Laryngeal involvement leads to decreased vocal pitch, progressive dyspnea, and stridor. Tracheal compression can contribute to dyspnea.29 Laryngoscopy shows edema and bulging of the false cords, with marked narrowing of the airway at this level, and difficulty in visualizing the true vocal cords. Radiography of the larynx shows a slit-like upper air passage and reduced vocal cord movement. Cervical fatty accumulation occurs in the anterior neck, and preepiglottic fat lies anterior to the larynx.30

Neck dissection, with extensive removal of huge lipomas, failed to relieve one patient initially.25 Laryngoscopic incision of a thin translucent false vocal cord mucosa released herniated fat, with slight improvement. A second open procedure via laryngofissure allowed excision of lipomatous tissue that bulged through the thyrohyoid membrane and dissected between thyroid laminae and laryngeal mucosa. Herniation of fat into the larynx extended from the anterior commissure to aryepiglottic folds posteriorly.

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