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 Table of Contents  
CASE REPORT
Year : 2016  |  Volume : 2  |  Issue : 1  |  Page : 115-118

A case of autopneumonectomy without any symptoms


Department of Respiratory, Critical Care and Sleep Medicine, Indraprastha Apollo Hospitals, New Delhi, India

Date of Submission01-Oct-2015
Date of Acceptance14-Nov-2015
Date of Web Publication2-Jun-2016

Correspondence Address:
Trilok Chand
B-241, Sarita Vihar, New Delhi - 110 076
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/2455-5568.183317

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  Abstract 

Autopneumonectomy simply describes the autolysis or almost complete destruction of the lung tissue without any surgical removal of the lung. This is not a common occurrence but is sometimes observed in areas where tuberculosis (TB) is endemic. The patients are usually diagnosed when they come to the hospital either with complications related to autopneumonectomy or a postpneumonectomy-like syndrome. Rarely, these patients remain asymptomatic until late age. Our case was an elderly female with an abnormal chest X-ray, who was referred to us for respiratory clearance for the surgical fixation of the fracture of the neck of femur. The history revealed that she had some chest infection and/or pulmonary TB in childhood that explained the autopneumonectomy. The patient underwent a successful surgery and was discharged with advice to follow-up for any chest complaints in the future.
The following core competencies are addressed in this article: Patient care, Medical knowledge.

Keywords: Autolysis, autopneumonectomy, mediastinal shifting, surgical pneumonectomy, tuberculosis


How to cite this article:
Chand T, Bansal A, Shankar V. A case of autopneumonectomy without any symptoms. Int J Acad Med 2016;2:115-8

How to cite this URL:
Chand T, Bansal A, Shankar V. A case of autopneumonectomy without any symptoms. Int J Acad Med [serial online] 2016 [cited 2019 Jan 17];2:115-8. Available from: http://www.ijam-web.org/text.asp?2016/2/1/115/183317


  Introduction Top


Autopneumonectomy is a term used to describe the almost complete destruction or vanishing of the lung secondary to the destructive lung disease. It is accompanied by compensatory enlargement of the other lung and mediastinal shifting toward the diseased side. The common destructive lung diseases, which produce autopneumonectomy are infections, commonly tuberculosis (TB) in the endemic areas, and other suppurative pulmonary diseases.[1],[2] These patients usually present with symptoms of obstructive lung disease. Nevertheless, this is less commonly associated than with the postpneumonectomy syndrome seen after surgical pneumonectomy.

Our patient is an elderly female who came to the hospital with a fractured hip and was accidentally diagnosed as a case of autopneumonectomy, as she never had any chest symptoms. A patient with autopneumonectomy from the Indian subcontinent, presenting without any pulmonary symptoms at the age of 81 years is a rare phenomenon.


  Case Report Top


An 81-year-old female presented in our hospital with a history of fall from the bed. She had sustained injury to the left hip and X-ray revealed an intertrochanteric fracture of the neck of the femur. All the routine investigations were done, and surgery was planned under general or spinal anesthesia.

The patient was referred to us for respiratory clearance for surgery with an abnormal chest X-ray (CXR). Her CXR showed an opaque right hemithorax with a shift of the mediastinum, heart, and trachea toward the right side [Figure 1]. On examination, she was short and of thin build with no apparent respiratory distress at rest. The oxygen saturation was 93% on room air. On auscultation, bilateral breath sounds were present, except right basal posteriorly, where breath sounds were decreased in intensity.
Figure 1: Chest X-ray shows opaque right hemithorax with mediastinal shifting

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The patient was posted for bronchoscopy in view of the suspected collapse of the right lung due to obstruction. During fibreoptic bronchoscopy, we found that there was neither any secretions nor any endobronchial growth, but all the bronchi distal to the right main bronchus were rudimentary and completely obliterated [Figure 2]. Her high resolution computed tomography (HRCT); of chest was ordered, which showed a shift of the mediastinum and the heart toward the right thoracic cavity. There was a small, tongue-shaped, degenerated, and calcified lung on the right side[Figure 3], which suggested autolysis or hypoplasia of the lung tissue due to some lung infection in childhood. Patient's two-dimensional echo showed mild left ventricular (LV) hypertrophy, good LV and right ventricular function, LV ejection fraction (LVEF)=60%, moderate pulmonary arterial hypertension (pulmonary artery systolic pressure = 53 mmHg), central venous pressure 10 mmHg, and dobutamine stress echo was negative for reversible ischemia (LVEF60–70%). Her ABG showed PH − 7.41, PaCo2 43.4 mmHg, PaO2 58.8 mmHg, and HCo3 was 26 mmHg. Patient's pulmonary function test (PFT) was not done in view of stable respiratory status and acceptable other parameters for surgery since the patient was clinically stable, so respiratory clearance was given, and she had undergone a successful surgery. The surgery was performed under spinal anesthesia (0.5% bupivacaine) and intravenous fentanyl, the patient was oxygenated by face mask in the right lateral position.
Figure 2: Fibreoprtic bronchoscopy shows rudimentary and obliterated right main bronchus

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Figure 3: High-resolution computed tomography chest showed small tongue-shaped degenerated and calcified right lung with extreme mediastinal shifting toward right side

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We reviewed the past medical history, which revealed that she had recurrent chest infections during her childhood, but neither was she evaluated nor treated properly. Since, the patient's parents are not alive, so the exact presentation of chest symptoms are not precisely mentioned by anyone of the family members, and we presumed that TB could be a reason for autopneumonectomy in this patient, because TB is so rampant in this region.


  Discussion Top


Autopneumonectomy refers to the functional destruction or under developement of the lungs due to destructive lung disease or infection, usually in childhood. As a compensatory mechanism for destruction or removal of the lobes or lung, the other lung and/or lobes overgrow and occupy the free space in the thoracic cavity. In adults, after autopneumonectomy in childhood, the single remaining lung has an alveolar number and surface area similar to that of the two lungs of a normal individual combined.[3] This happens in early childhood, typically by the age of 8 years,[4] because in mammals alveolar multiplication ceases a few years after birth. Some studies also showed that immature animals respond to surgical pneumonectomy with compensatory lung growth, while most adult mammals do not.[5] Evidence of compensatory lung growth in humans has been found in children who underwent lobar resections for congenital lobar emphysema during the 1st year of life.[6]

The true incidence of autopneumonectomy by any cause is still unknown, and only limited literature available on autopneumonectomy. The absence of lung tissue in the thoracic cavity since childhood may be due to congenital pulmonary hypoplasia or autopneumonectomy as a result of infection. The unilateral pulmonary hypoplasia occurs on the right side much more often than on the left side and is usually associated with abnormalities of the main stem bronchus.[7] The common etiologic agent for autopneumonectomy is childhood pulmonary infection with mycobacterium tuberculosis, which is common worldwide. Pulmonary involvement of the infective agent mycobacterium tuberculosis can lead to bronchiectasis, and ultimately pulmonary destruction and fibrosis, if left untreated.[1],[2] Other infections, such as Streptococcus pneumoniae, Haemophilus influenzae, Staphylococcus aureus and Mycoplasma pneumonia in their severe form, lead to gangrenous lung or autopneumonectomy. The Klebsiella infection is well-known pathogen for necrotizing pneumonia and extensive tissue destruction, which also cause autopneumonectomy. The autopneumonectomy by all these community-acquired infections is also termed as “dry gangrene,” in which affected lung becomes functionless and prevent further systemic spread of infection.[8] These infections cause autopneumonectomy by severe inflammation of the lung, exudation and ventilation-perfusion mismatch, necrosis, followed by abscess formation, and gangrenous changes. In pulmonary TB, mycobacterium infection causes granuloma formation, necrosis, and cavitation and further tissue destruction at the edges of the cavity.

These patients usually remain asymptomatic until elderly life, or they may develop some obstructive changes in lung functions. In the early childhood or adolescent period, they may present with intermittent hemoptysis.[9] The elderly or older age patients usually present with some degree of dyspnea due to a progressive decline in lung function with ageing, single functioning lung, or pulmonary infections in the lifetime. Though, postpneumonectomy-like syndrome is uncommon after functional pneumonectomy secondary to destructive lung disease.[10],[11],[12] Interestingly, the post pneumonectomy-like syndrome is more commonly seen with right-sided lung resection, due to the counterclockwise rotation of the mediastinum into the pneumonectomy space.[1] As the same mechanism, patients with right-sided autopneumonectomy must have more symptoms, which has not happened in our case. When no surgical intervention is involved, a postpneumonectomy-like syndrome has been described, where the destructive lung disease is the etiological agent, rather than surgical resection of the lung.[1],[13]

Our case was an 81-year-old female who had never complained of any chest symptoms in her adult as well as elderly life, except a vague history of recurrent chest infections in early childhood. At this age, it is very uncommon that a patient with only a single functioning lung remains asymptomatic. This happened in our case, probably because the other lung of the patient had overgrown completely. It structurally appeared normal, and there was no evidence of airway obstruction or bronchial compression.

The diagnosis of autopneumonectomy is based on compatible history and radiological findings. These patients and/or sometimes their parents give the history of recurrent chest infection or pulmonary TB in infancy or childhood. Most of the patients present in hospital with some pulmonary complications, and CXRs or CT-chest revealed an extensive shift of mediastinum, overgrowth of the contralateral lung and destruction or fibrosis of ipsilateral lung. However, few patients remain asymptomatic until late age. They are incidentally found to have autopneumonectomy when evaluated for some other medical reason, as in our case. The PFT sometimes shows the normal result or sometimes obstructive changes with raised residual volume and reduction of the maximum expiratory flow.[6],[14] The autopneumonectomy patients really do not require any specific treatment until and unless there are any complications. In this situation, we recommend only conservative treatment. If a patient with autopneumonectomy has compromised respiratory status, referral for pulmonary rehabilitation is warranted. Other management approaches include prevention of further infections, rapid diagnosis, and treatment of postpneumonectomy-like syndrome, though it is a very rare instance in the case of autopneumonectomy.


  Conclusion Top


Autopneumonectomy is not a common clinical condition, and most of the patients present with some complication. Autopneumonectomy is a diagnosis of exclusion where no other etiology can explain unilateral hypoplasia of the lung. Any patient, who has a history of recurrent chest infections or suspected pulmonary TB in infancy or childhood, should be evaluated by PFTs and radiological imagings, because a timely diagnosis of autopneumonectomy can help us to prevent future complications and morbidity.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
  References Top

1.
King J, Lau RW, Wan I, Yim A. Tuberculosis tracheobronchial stricture causing postpneumonectomy-like syndrome corrected by insertion of a bespoke Dumon stent. Int Cardiovasc Thorac Surg 2008;7:267-8.  Back to cited text no. 1
    
2.
Kashyap S, Mohapatra PR, Saini V. Endobronchial tuberculosis. Indian J Chest Dis Allied Sci 2003;45:247-56.  Back to cited text no. 2
    
3.
Thurlbeck WM, Galaugher W, Mathers J. Adaptive response to pneumonectomy in puppies. Thorax 1981;36:424-7.  Back to cited text no. 3
    
4.
Thurlbeck WM. Postnatal human lung growth. Thorax 1982;37:564-71.  Back to cited text no. 4
    
5.
Thurlbeck WM. Postnatal growth and development of the lung. Am Rev Respir Dis 1975;111:803-44.  Back to cited text no. 5
    
6.
McBride JT, Wohl ME, Strieder DJ, Jackson AC, Morton JR, Zwerdling RG, et al. Lung growth and airway function after lobectomy in infancy for congenital lobar emphysema. J Clin Invest 1980;66:962-70.  Back to cited text no. 6
    
7.
Krummel TM. Congenital malformations of the lower respiratory tract. In: Chernick V, Boat TF, editors. Kendig's Disorders of the Respiratory Tract in Children. 6th ed. Philadelphia: WB Saunders; 1998. p. 307-8.  Back to cited text no. 7
    
8.
Chih-Hao Chen (2011). Gangrenous Lung Disease, Gangrene-Current Concepts and Management Options, Dr. Alexander Vitin (Ed.), ISBN: 978-953-307-386-6  Back to cited text no. 8
    
9.
Ciric I, Meyers MP, Mayba J, Anthonisen NR. Autopneumonectomy with compensatory lung growth. Can Respir J 2003;10:271-3.  Back to cited text no. 9
    
10.
Grillo HC, Shepard JA, Mathisen DJ, Kanarek DJ. Postpneumonectomy syndrome: Diagnosis, management, and results. Ann Thorac Surg 1992;54:638-50.  Back to cited text no. 10
    
11.
Valji AM, Maziak DE, Shamji FM, Matzinger FR. Postpneumonectomy syndrome: Recognition and management. Chest 1998;114:1766-9.  Back to cited text no. 11
    
12.
Furia S, Biban P, Benedetti M, Terzi A, Soffiati M, Calabrò F. Postpneumonectomy-like syndrome in an infant with right lung agenesis and left main bronchus hypoplasia. Ann Thorac Surg 2009;87:e43-5.  Back to cited text no. 12
    
13.
Veronesi G, Spaggiari L, Solli PG, Leo F, Villa G, Pastorino U. Postpneumonectomy-like syndrome after chemoradiation therapy for lymphoma. Ann Oncol 2002;13:1945-7.  Back to cited text no. 13
    
14.
Ford GT, Galaugher W, Forkert L, Fleetham JA, Thurlbeck WM, Anthonisen NR. Static lung function in puppies after pneumonectomy. J Appl Physiol Respir Environ Exerc Physiol 1981;50:1146-50.  Back to cited text no. 14
    


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  [Figure 1], [Figure 2], [Figure 3]



 

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