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Ann Thorac Surg 1997;64:226-228
© 1997 The Society of Thoracic Surgeons

Case Report

Unilateral Giant Bullous Emphysema With Placental Transmogrification of the Lung

W. Stewart Horsley, MD, Anthony A. Gal, MD, Kamal A. Mansour, MD

Division of Cardiothoracic Surgery, Joseph P. Whitehead Department of Surgery, and Department of Pathology, Emory University School of Medicine, Atlanta, Georgia

Accepted for publication February 13, 1997.


   Abstract
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Introduction
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A case of severe unilateral bullous emphysema in an otherwise healthy young man is presented to highlight a rare histologic variant termed "placental transmogrification of the lung." The etiology is unknown, but operation has been curative in the small number of cases reported.


   Introduction
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Bullous emphysema presents typically with diffuse bilateral lung involvement in older individuals with a long smoking history. Uncommon causes such as {alpha}1-antitrypsin deficiency usually also manifest bilaterally and progress slowly over time. The following case report describes a patient with a rare histologic variant of unilateral giant bullous emphysema involving placental transmogrification of the lung. This entity is very unusual from both a clinical and pathologic standpoint, with only a few cases reported in the pathology literature [1–3].

The patient was a 28-yar-old Mexican-American male construction worker who had a 3-month history of cough, including scant hemoptysis, left-sided pleuritic chest pain, and gradual shortness of breath. There was a vague history of an antecedent upper respiratory viral infection, but otherwise he was in excellent health. He smoked cigarettes only occasionally in the distant past but not during the previous 6 years. He had pneumonia 2 years previously but did not require hospitalization.

The physical examination was remarkable for a significant tracheal shift to the right and almost complete absence of breath sounds on the left. Heart tones were auscultated over the right hemithorax. Other than these findings, he appeared healthy and robust.

The initial chest roentgenogram showed large unilateral bullae throughout the left lung with marked pulmonary overinflation and considerable mediastinal shift to the right (Fig 1Go). This shift compressed his right lung, which otherwise appeared normal. A chest computed tomographic scan confirmed these findings, showing diffuse emphysematous appearing bullae on the left, with little if any normal left lung and no mass lesions. Fiberoptic bronchoscopy showed some luminal narrowing and mild inflammatory changes in the left upper lobe and left lower lobe posterior basal bronchi. Bronchial washings for bacterial and mycobacterial cultures were negative. A battery of laboratory tests including a complete blood work, electrolytes, liver function studies, and a skin purified protein derivative of tuberculin test were normal. A serum {alpha}1-antitrypsin level was not obtained. Quantitative ventilation-perfusion lung scanning showed near-total preferential ventilation and perfusion to the right lung. Preoperative pulmonary function tests were consistent with an obstructive pattern (forced expiratory volume in 1 second, 1.71 L; 45% predicted).



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  		Fig 1. . Chest roentgenogram shows unilateral diffuse emphysematous bullae on the left side with marked mediastinal shift to the right.

 
Because of the severe unilateral nature of the bullae, with essentially no functioning left lung and mediastinal compression of the right lung, a left pneumonectomy was done through a left thoracotomy. The patient's breathing improved significantly postoperatively, and follow-up pulmonary function test results 2 months after the operation were slightly improved (forced expiratory volume in 1 second, 2.21 L; 52% predicted). The mediastinum shifted back to the midline as seen on follow-up chest roentgenograms (Fig 2Go).



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  		Fig 2. . Chest roentgenograms 4 days (A) and 5 weeks (B) postoperatively show progressive centralization of the mediastinum and good expansion of the right lung.

 
On gross examination, the left lung was enlarged, measuring 27 x 20 x 2.5 cm, and weighed 840 g. It contained partially collapsed emphysematous bullae that ranged from 0.5 to 8.0 cm. The lower lobe was more homogeneous, velvety, and brown-red and lacked the bullous emphysematous changes seen in the upper lobe.

The microscopic appearance of the lung was remarkable for its diverse histologic patterns. In the upper lobe, there were large, thin-walled emphysematous bullae. Scattered, peculiar eosinophilic, myxoid villous structures, associated with rare chronic inflammatory cells, were present within the bullae, but not elsewhere in the lung (Fig 3Go). These lesions had a striking resemblance to placental chorionic villi (Fig 4Go), but there was no immunostaining for ß-human chorionic gondaotropin (Biogenex, San Roman, CA; 1:1600) or placental alkaline phosphatase (Dako Corp, Carpenteria, CA; 1:800). In addition, there were small foci of papillary, irregular structures containing arborizing blood vessels. The lower lobe lacked the villous or papillary structures, but showed prominent interstitial thickening. This was represented by an anastomosing thick network of bland stromal cells intertwined with capillaries. A Mallory's trichrome stain did not show any increased interstitial collagen, but a Verheoff-Van Gieson stain showed slightly increased elastic staining surrounding groups of bland stromal cells. The actin immunostain (anti-{alpha}-actin; Bioteck Solutions, Santa Barbara, CA; no dilution) was markedly positive within the interstitium accentuating small capillaries.



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  		Fig 3. . Emphysematous bullae with myxoid villous structures. (Hematoxylin and eosin; x10 before 50% reduction.)

 


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  		Fig 4. . The edematous, myxoid villous structures resemble placental chorionic villi. (Hematoxylin and eosin; x50 before 50% reduction.)

 

   Comment
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 Abstract
 Introduction
 Comment
References
 
Severe unilateral bullous emphysema with diffuse destruction of a single lung is quite uncommon. The predominant clinical forms of emphysema, such as those due to smoking or {alpha}1-antitrypsin deficiency, usually affect both lungs to some degree. The most striking histologic feature in our patient was the peculiar placental-like papillary and villous structures associated with a cellular interstitial stroma resembling chorionic villi that has been termed "placental transmogrification." This form of emphysema was first reported in an abstract in 1979 [1] and recently has been described in two articles in the pathology literature with a total of 7 patients [2, 3]. These 7 patients consisted of 5 men, ages 24, 27, 27, 38, and 44 years, and 2 women, ages 28 and 33 years. Five patients had lobectomy and 2 had pneumonectomy, although it is not clear from the reports whether the patients who only had lobectomy had disease confined to a single lobe. At least 1 patient who underwent lobectomy later was found on computed tomographic scan to have emphysematous changes in the remaining ipsilateral lobe. Despite the few patients reported, all have done well after pulmonary resection, with a follow-up range of from 1 to 10 years. Two patients had a history of pneumonia as a child, and 1 had cerebral palsy.

The patient we presented is typical of previously reported cases. The slight improvement in the postoperative forced expiratory volume in 1 second after pneumonectomy indicates that the resected lung was so diseased that it was essentially nonfunctional. This improvement is probably related to a better ventilation-perfusion match in the remaining right lung.

Placental transmogrification of the lung should be in the differential diagnosis of any patient who presents with predominantly unilateral bullous disease, especially in those without other risk factors for emphysema. The etiology of this rare disease is unknown, but surgical resection has been curative in the small number of patients so far reported.


   Footnotes
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Address reprint requests to Dr Mansour, The Emory Clinic, 1365 Clifton Rd, Atlanta, GA 30322.


   References
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 Footnotes
 Abstract
 Introduction
 Comment
 References
 

  1. McChesney T. Placental transmogrification of the lung: a unique case with remarkable histopathologic features [Abstract]. Lab Invest 1979;40:245–6.
  2. Fidler ME, Koomen M, Sebek B, Greco MA, Rizk CC, Askin MB. Placental transmogrification of the lung, a histologic variant of giant bullous emphysema: clinicopathological study of three further cases. Am J Surg Pathol 1995;19:563–70.[Medline]
  3. Mark EJ, Muller KM, McChesney T, Dong-Hwan S, Honig C, Mark MA. Placentoid bullous lesion of the lung. Hum Pathol 1995;26:74–9.[Medline]



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This Article
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Right arrow Articles by Mansour, K. A.

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