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Ann Thorac Surg 1998;66:566-567
© 1998 The Society of Thoracic Surgeons

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Case Reports

Infection with Mycobacterium tuberculosis complicating a pulmonary sequestration

^a Department of Thoracic Surgery, University "Federico II", Naples, Italy
^b Department of Pathology, University "Federico II", Naples, Italy

Accepted for publication March 3, 1998.

Address reprint requests to Dr Alifano, Via Roma 60, I-81024 Maddaloni, Italy

	Abstract

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Pulmonary sequestration is a relatively rare malformation. Infection with common pyogenes is a frequent feature in the evolution of this disease. We report a case of intralobar sequestration infected with Mycobacterium tuberculosis in the absence of any other site of tuberculous infection. The patient underwent surgical removal of the affected lobe and subsequent antituberculous chemotherapy. At 1-year follow-up his clinical status is excellent.

	Introduction

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Pulmonary sequestration is a relatively uncommon congenital disease. Although respiratory distress (especially in the newborn or the infant), intrapleural hemorrhage, or congestive heart failure secondary to left-to-right shunt may represent the revealing symptoms [1–3], repeated bacterial infections are the most frequent modality of presentation of the disease.

We report herein a case of intralobar sequestration complicated by infection with Mycobacterium tuberculosis.

A 61-year-old man presented with a 3-week history of cough, malaise, and fever (>38°C). Moderate (4 kg) weight loss was also reported. He was a current heavy smoker (40 packs/year). His past medical history included only a peptic ulcer treated with anti-H₂ drugs. On admission he had no respiratory distress. Pulse was regular at 78 beats/min and blood pressure was 140/80 mm Hg. Physical examination revealed attenuation of respiratory murmur at the left basal area. There was neither cyanosis nor digital clubbing. With the patient breathing room air, arterial blood-gas analysis showed a pH of 7.40, an arterial carbon dioxide tension of 37 mm Hg, and an arterial oxygen tension of 77 mm Hg. Blood laboratory studies disclosed no further abnormalities. Results of human immunodeficiency virus testing were negative. Chest radiography and computed tomographic scanning revealed a left-sided basal gross opacity (Fig 1). A computed tomography-guided fine-needle aspiration was nondiagnostic. Although the presence of a pulmonary sequestration was hypothesized because of the topography of the lesion, aortography was not performed because it was unavailable at that time.

View larger version (98K): [in this window] [in a new window]   Fig 1. Computed tomographic scan of the thorax showing a gross opacity in the left lower lobe.

View larger version (140K): [in this window] [in a new window]   Fig 2. Histologic slide showing lung parenchyma with evidence of atelectasis and congestion. Caseating granulomata and giant cells are also present. (Hematoxylin and eosin; x106 before 46% reduction.)

	Comment

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Pulmonary sequestration is a congenital malformation characterized by the presence of lung tissue with abnormal or absent communication with the bronchial tree and arterial blood supply from the systemic circulation. A sequestration within the visceral pleura is classified as intralobar sequestration (approximately 75% of cases), whereas a sequestration outside the visceral pleura is classified as extralobar sequestration. Intralobar sequestration affects the lower lobes in almost all the cases (98%), the left side being more frequently involved. The malformation may cause respiratory distress or other respiratory symptoms in the first days or months of life [1, 3] or it may remain asymptomatic for several years. Fifteen percent of patients remain symptom-free for all their life, and in these cases normal longevity is reported [3, 6]. However, in the majority of cases, repeated pulmonary infections occur. Minor hemoptysis is a frequent symptom; massive hemoptysis represents an unusual event that may be fatal [7].

Infections are generally caused by pyogenes, although fungal infections have been reported [8]. In our case M tuberculosis was responsible for the infection, in the absence of any other evident site of pulmonary or extrapulmonary tuberculosis. Mycobacterium tuberculosis might have reached the sequestration through Kohn’s pore (there was no evidence of anomalous bronchi) without causing evident disease in the normal lung parenchyma. However, we cannot exclude a hematogenous route of the tuberculous infection via lymphatics as the result of a progressive primary infection. Our patient presented with systemic symptoms related to tuberculous infection (fever, weight loss, malaise). Such symptoms disappeared after surgical resection and antituberculous chemotherapy. Similarly, titers of antimycobacterial antibodies returned to baseline 1 year after the intervention, suggesting the complete control of the tuberculous infection [4, 5].

Infection with M tuberculosis may complicate a pulmonary sequestration in the absence of any other apparent site of tuberculous infection. Surgical resection allows establishment of the exact diagnosis as well as immediate removal of the infectious focus, thus preventing the complications related to the infection or to the malformation itself.

	References

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1. Khalil K.G., Kuman J.W. Pulmonary sequestration. J Thorac Cardiovasc Surg 1975;70:928-937.[Abstract]
2. Zumbro G.L., Green D.C., Brott W., Treasure R.L. Pulmonary sequestration with spontaneous intrapleural hemorrhage. J Thorac Cardiovasc Surg 1974;68:673-674.[Medline]
3. Ryckman F.C., Rosenkrantz J.G. Thoracic surgical problems in infancy and childhood. Surg Clin North Am 1985;65:1423-1454.[Medline]
4. Alifano M., Sofia M., Mormile M., et al. IgA immune response against the mycobacterial antigen A60 in patients with active pulmonary tuberculosis. Respiration 1996;63:292-297.[Medline]
5. Alifano M., De Pascalis R., Sofia M., Faraone S., Del Pezzo M., Covelli I. Evaluation of IgA-mediated humoral immune response against the mycobacterial antigen P-90 in diagnosis of pulmonary tuberculosis. Chest 1997;111:601-605.[Abstract/Free Full Text]
6. Savic B., Birtel F.J., Tholen W., Funke H.D., Knoche R. Lung sequestration: report of seven cases and review of 540 published cases. Thorax 1979;34:96-101.[Abstract/Free Full Text]
7. Rubin E.M., Garcia H., Horowitz M.D., Guerra J.J., Jr Fatal massive hemoptysis secondary to intralobar sequestration. Chest 1994;106:954-955.[Abstract/Free Full Text]
8. Tanaka M., Miyamoto H., Sakao Y., Harada R., Hata F. A case report of intralobar sequestration associated with lung aspergillus. Nippon Kyobu Geka Gekkai Zasshi 1995;43:366-370.

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