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Ann Thorac Surg 1996;62:990-993
© 1996 The Society of Thoracic Surgeons


Original Articles: General Thoracic

Resection of the Right Middle Lobe and Lingula for Mycobacterial Infection

Marvin Pomerantz, MD, James R. Denton, MD, Gwen A. Huitt, MD, James M. Brown, MD, Lorie A. Powell, ANP, Michael D. Iseman, MD

Division of Cardiothoracic Surgery, Department of Surgery, University of Colorado Health Sciences Center, and Clinical Mycobacterial Service, Division of Infectious Disease in the Department of Medicine, The National Jewish Center for Immunology and Respiratory Medicine, Denver, Colorado


    Abstract
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Background. In a series of 229 patients infected with mycobacterial organisms, we noted a specific female phenotype that involves isolated infections of the middle lobe and lingula.

Methods. Thirteen patients were found to have infections of the middle lobe, lingula, or both. All of them were infected with Mycobacterium other then Mycobacterium tuberculosis, all were women, 12 of the 13 were slender, and most had variable combinations of skeletal abnormalities. All underwent resection of the middle lobe, lingula, or both.

Results. There were no operative deaths. Only 2 patients have had reactivation requiring additional antibiotic therapy. All patients have had a decreased number of pulmonary infections in the postoperative period. Anatomic findings at operation included a complete major fissure and at least a partially complete minor fissure with middle lobe resections or an elongated lingula.

Conclusions. Mycobacterial infection of the middle lobe and lingula is primarily a disease of asthenic women and is often associated with skeletal abnormalities and complete fissures or an elongated lingula. We recommend that surgical intervention be performed early once the condition is identified.


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See also page 993.

From a large group of Mycobacterium-infected patients, a subgroup has been noted who primarily have infection of the right middle lobe, lingula, or both. Right middle lobe and lingular infection has long been recognized as a unique clinical syndrome [1]. Its association with mycobacterial infection has been more recent. The mechanism of obstruction to a single lobar bronchus by enlarged peribronchial lymph nodes was thought to lead to chronic infection, bronchiectasis, and eventual destruction of the middle lobe, lingula, or both [2, 3]. However, a complex mechanism is more likely responsible for this isolated condition. Mycobacterium other than Mycobacterium tuberculosis (MOTT) has recently been recognized as a frequent and increasing pathogen in both immunosuppressed and nonimmunosuppressed patients [46]. Our series and other recent series [4, 7] document isolated middle lobe and lingular involvement in nonimmunosuppressed patients infected with MOTT. A specific phenotype emerges when this group of patients is examined. The purpose of this report is to analyze the specific characteristics of patients with disease of the middle lobe, lingula, or both who are infected with MOTT organisms, to review pathogenic theories, and to document the results of surgical resection.


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Between August 1983 and January 1996, our group operated on 229 patients with mycobacterial infections. Of these patients, 123 had multidrug-resistant Mycobacterium tuberculosis, 96 had MOTT, and 10 had nonresistant Mycobacterium tuberculosis infections. Organisms were specifically identified by culture from either the sputum or bronchoscopy.

Of the 96 patients with MOTT infections, 13 had isolated infections of the right middle lobe, lingula, or both. Nine of the 13 patients were operated on for middle lobe disease (Fig 1Go), 2 were operated on for lingular disease (Fig 2Go), and the remaining 2 had both the middle lobe and lingula resected for bilateral mycobacterial infection (Table 1Go). Age at the time of operation ranged between 45 and 70 years, and 8 patients were in their 50s. All 13 patients were women. One patient had a long history of reflux and pulmonary infections as a child. The remaining 12 were slender, and most had variable combinations of skeletal abnormalities consisting of pectus excavatum, scoliosis, or a straight back. A number of these women also had mitral prolapse.



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Fig 1. . (A) Computed tomographic scan in patient with Mycobacterium avium infection of middle lobe. (B) High-resolution scan of middle lobe disease.

 


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Fig 2. . Computed tomographic scan in patient with Mycobacterium avium infection of lingula.

 

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Table 1. . Summary of Patient Data
 
All 13 patients had been on a regimen of antimicrobial therapy for long periods, sometimes up to 15 years, prior to specific treatment of their mycobacterial disease. The infecting organism was Mycobacterium avium/intracellulare complex in 12 and Mycobacterium chelonae in 1. The decision to operate was based on the chronicity of the disease, the persistence of hemoptysis, and the presence of severe bronchiectasis or an entirely destroyed lobe with or without positive sputum. Surgical intervention was performed after patients had been on a regimen of drug-specific therapy for about 3 months.

The surgical approach involved use of a double-lumen endobronchial tube. A lateral thoracotomy incision sparing the anterior serratus muscle was used in all patients. At operation, it was noted that with middle lobe disease, the major fissure was always complete and the minor fissure, at least partially complete. The lingula was unremarkable except for what appeared to be an elongated tip. No other specific anatomic findings were evident. In 6 of the 13 patients, milky-white secretions were seen emanating from the infected bronchus at bronchoscopy, routinely performed prior to thoracotomy. In view of this finding, patients seen since 1993 have had bronchoscopy at the completion of operation to remove any secretions present in the trachea or bronchi after surgical manipulation. No instance of total bronchial obstruction was noted at the time of operation. Lobectomy, lingulectomy, or both were performed without use of a muscle flap even if the sputum was positive for Mycobacterium at the time of operation. It was not thought necessary to use muscle flaps, as the bronchial stump was rapidly covered by the residual lung, and that probably acted as a buttress to the suture line closure.

Postoperative care was standard and, as in most pulmonary resections, included early mobilization and aggressive respiratory care. Culture-specific antibiotics were continued for 18 to 24 months postoperatively unless poorly tolerated by the patient. In these instances, antibiotics were usually stopped 12 months postoperatively. Follow-up has included serial roentgenograms, sputum cultures, and review of the clinical course.


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There were no operative deaths, and all patients were alive at the time this report was written. There were no intraoperative complications, although 1 patient required tube thoracostomy for recurrent pneumothorax after the initial chest tubes had been removed. The majority of patients were discharged 5 days postoperatively and usually returned to National Jewish Hospital for physical therapy and readjustment of medications. All patients have been followed either by physicians at National Jewish Hospital or by the referring physician, who remains in contact with physicians at National Jewish Hospital.

All patients are either completely well or have had far fewer pulmonary infections postoperatively than preoperatively. All had more energy and a better state of well-being. The results for each patient are included in Table 1Go. Six patients are in stable condition without medication. Four patients have had Pseudomonas pulmonary infections during follow-up, and 2 patients are doing well early in the postoperative course but remain on a regimen of antimycobacterial medication. One patient infected with Mycobacterium avium/intracellulare complex experienced reactivation several years after operation, was treated again with antimycobacterial therapy, and is currently well. The 1 patient infected with Mycobacterium chelonae did well for 4 years. However, she recently has had symptoms and is infected with Mycobacterium avium and shortly will be started on a regimen of medication.


    Comment
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Nontuberculous mycobacterial infection has long been recognized as an enigmatic problem sometimes resistant to medical therapy. This series as well as others recognizes a specific pattern of disease characterized initially by isolated involvement of the middle lobe, the lingula, or both. A phenotype consisting of thin women with thoracic skeletal abnormalities has been found to be associated with this pattern of middle lobe and lingular disease. The operative findings of complete major fissure and partially complete minor fissures is also characteristic. Iseman and associates [8] have made similar observations on the association of pectus excavatum and scoliosis with these Mycobacterium avium/intracellulare complex infections. The cause of isolated lingular involvement remains less clear but may be related to an elongated lingula as well as the specific phenotype.

The pathogenic mechanism by which this particular phenotype predisposes these patients to chronic, unrelenting infections remains unclear. Reisch and Johnson [7] theorized that voluntary suppression of the cough reflex in fastidious women predisposes to chronic infection and used the term Lady Windemere syndrome for this disease pattern. Others [8] offer defects at the molecular level as an explanation for the syndrome. The coexistence of thoracic skeletal abnormalities, pulmonary destruction, and mitral valve prolapse has led one of us [9] to classify this syndrome as a connective tissue disorder. Complete and partially complete fissures isolate these areas of the lung from drainage by way of collateral ventilation. This finding was observed by Bradham and colleagues [10] in 1966 and postulated to be a contributory factor to chronic middle lobe infections. The anatomic findings in this series tend to support this theory. The lack of collateral ventilation combined with tissue weakness at the cellular level would certainly predispose these patients to chronic infections. Thoracic skeletal abnormalities resulting in decreased cough and sputum clearance may also contribute to progression of the disease. The true pathogenic mechanism is most likely multifactorial and will require further investigation.

In summary, MOTT infections of the middle lobe and lingula appear to be a disease of women with a specific phenotype. In general, these women are asthenic and have a variety of skeletal abnormalities. At the time of operation, there is a complete major fissure and at least a partially complete minor fissure when the middle lobe is resected. The etiology of the lingular disease is not as clear but may be related to an elongated lingula as well as the described phenotype. We believe that these patients do better with surgical intervention combined with appropriate antibiotic therapy. In our series, they either were cured or had a decreased number of pulmonary infections. On the basis of our experience, we recommend that operation be performed early in patients with isolated MOTT infections of the middle lobe, lingula, or both before other portions of the lung become grossly diseased and are more likely to be susceptible to subsequent infection. It is important to continue antibiotics for 12 to 24 months postoperatively, and patients need continued follow-up to treat either recurrence of mycobacterial disease or superinfection in other portions of the lung.


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Presented at the Thirty-second Annual Meeting of The Society of Thoracic Surgeons, Orlando, FL, Jan 29–31, 1996.

Address reprint requests to Dr Pomerantz, Division of Cardiothoracic Surgery, University of Colorado Health Sciences Center, 4200 E 9th Avenue, Box C310, Denver, CO 80262.


    References
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  1. Graham EA, Burford TH, Mayer JH. Middle lobe syndrome. Postgrad Med 1948;4:29–34.[Medline]
  2. Brock RC, Conn RJ, Dickinson JR. Tuberculous mediastinal lymphadenitis in childhood: secondary effects on lungs. Guy's Hosp Rep 1937;87:295–317.
  3. Brock RC. Post tuberculous broncho-stenosis and bronchiectasis of the middle lobe. Thorax 1950;5:5–39.[Free Full Text]
  4. Kennedy TP, Weber DJ. Nontuberculous mycobacteria: an underappreciated cause of geriatric lung disease. Am J Respir Crit Care Med 1994;149:1654–8.[Abstract]
  5. Miller B, Faber LP, Snider DE. Mycobacteriosis and the acquired immunodeficiency syndrome. Curr Pulm 1987;8:357–70.
  6. Prince DS, Peterson DD. Infection with Mycobacterium avium complex in patients without predisposing conditions. N Engl J Med 1989;321:863–8.[Abstract]
  7. Reich JM, Johnson RE. Mycobacterium avium complex pulmonary disease presenting as an isolated lingular or middle lobe pattern: the Lady Windermere syndrome. Chest 1992;101:1605–9.[Abstract/Free Full Text]
  8. Iseman MD, Buschman DL, Ackerson LM. Pectus excavatum and scoliosis: thoracic abnormalities associated with pulmonary disease caused by Mycobacterium avium complex. Am Rev Respir Dis 1991;144:914–6.[Medline]
  9. Iseman MD. "That's no lady" [Letter]. Chest (in press).
  10. Bradham RR, Sealy WC, Young WG Jr. Chronic middle lobe infection: factors responsible for its development. Ann Thorac Surg 1966;2:612–6.[Medline]

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Ann. Thorac. Surg. 1996 62: 993. [Extract] [Full Text]



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