Ann Thorac Surg 2009;88:287-288. doi:10.1016/j.athoracsur.2008.12.031
© 2009 The Society of Thoracic Surgeons
Case Reports
Severe Hypoxemia Due to Intrapulmonary Shunting Requiring Surgery for Bronchioloalveolar Carcinoma
Pierre-Emmanuel Falcoz, MD, PhDa,*,
Nhum Tran Khai Hoan, MDb,
Françoise Le Pimpec-Barthes, MD, PhDb,
Marc Riquet, MD, PhDb
a Department of Thoracic Surgery, University Hospital, Strasbourg, France
b Department of Thoracic Surgery, Georges Pompidou European Hospital, Paris, France
Accepted for publication December 9, 2008.
* Address correspondence to Dr Falcoz, Department of Thoracic Surgery, Nouvel Hôpital Civil, Hôpitaux Universitaires de Strasbourg, 1 place de l'Hôpital, Strasbourg Cedex, BP 426, 67091, France (Email: pierre-emmanuel.falcoz{at}wanadoo.fr).
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Abstract
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Bronchioloalveolar carcinoma is a rare, but well-known disease that symptomatically worsens with intrapulmonary shunting and consequent hypoxemia. Surgical resection of the involved area offers relief from disabling hypoxemia and may improve survival. We present 3 patients with intrapulmonary shunting.
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Introduction
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Bronchioloalveolar carcinoma (BAC) of the lung is a subtype of adenocarcinoma with a pure bronchoalveolar growth pattern and no evidence of stromal, vascular, or pleural invasion (World Health Organization criteria). We report 3 patients with BAC and severe hypoxemia, due to intrapulmonary shunting, managed surgically.
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Case Reports
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Patient 1
A 68-year-old man was admitted for surgical management of a middle lobe tumor. On computed tomographic (CT) scanning, an opacified right middle lobe with lesser similar lesions in the right upper and lower lobes were noted. Cytologic studies of bronchioloalveolar lavage supported the diagnosis of BAC. Spirometry showed a decrease in forced expiratory volume in 1 second to 1.91 L (71% of predicted). Arterial blood gas analysis on room air showed severe hypoxemia with a pH of 7.45, PaCO
2 of 33.0 mm Hg, and PaO
2 of 45.0 mm Hg, indicative of an intrapulmonary shunt. Blood gases improved when the right pulmonary artery was occluded with a balloon catheter. These findings suggested that pulmonary resection would relieve hypoxia. A middle lobectomy and resection of the anterior segment of the upper lobe was well tolerated. Arterial blood gas analysis on room air improved dramatically (pH 7.43; PaCO
2, 41 mm Hg; PaO
2, 72 mm Hg). The patient was discharged on postoperative day 7. Unfortunately, despite chemotherapy, recurrence of disease bilaterally was evident at 18 months, with death occurring at 24 months.
Patient 2
A 54-year-old man was evaluated for severe dyspnea, which required 3 L/minute of supplemental oxygen and bronchorrhea. On CT scan, diffuse ground glass opacification of the left upper lobe, most of the lower lobe, and scattered areas in the right lower lobe were present. Spirometry showed a decrease in forced expiratory volume in 1 second to 1.68 L (57% of predicted) and forced vital capacity (FVC) to 2.37 L (65% of predicted). Arterial blood gas analysis on room air showed hypoxemia with a pH of 7.45, a PaCO2 of 33.0 mm Hg, and a PaO2 57.0 of mm Hg. A left pulmonary biopsy was done by video-assisted surgery, which indicated BAC. The patient was enrolled on a lung transplant list. Due to decreasing tolerance, presumed because of the functional intrapulmonary shunt (increasing supplementation in O2), a left pneumonectomy associated with radical systematic lymphadenectomy was performed with immediate satisfactory results. Arterial blood gas analysis on room air improved (pH 7.42; PaCO
2, 39 mmHg; PaO
2, 75 mm Hg). Oncological follow-up showed no evidence of residual or recurrent disease, and the patient was maintained on the transplant list. A right lung transplantation was performed 16 months later. The patient died postoperatively due to colic perforation.
Patient 3
A 63-year-old man was hospitalized and successfully treated for left lower lobe pneumonia. He was hospitalized again 5 months later for cough and hemoptysia. A CT scan revealed a homogenous alveolar consolidation of the left lower lobe associated with ground glass opacities of the right upper lobe. Bronchoscopy revealed no endobronchial lesions; transbronchial biopsies showed histologic features consistent with BAC. The patient received six regimens of platinum-based chemotherapy during an 8-month period, followed by Erlotinib due to clinical evolution of the cancer. He was readmitted for acute aggravation of dyspnea and left pneumothorax. His respiratory condition remained precarious despite thoracic drainage. Arterial blood gas values, with the patient breathing 15 L/minute of O2, showed a pH of 7.44, a PaCO
2 of 38.0 mm Hg, a PaO
2 of 55.5 mm Hg, and an oxygen saturation value of 87.8%. An angio-CT scan showed alveolar condensation of the entire left lower lobe with an angiogram sign associated with infiltrative lesion of the lingula, and right upper and lower lobes. There were no signs in favor of pulmonary embolism. An arteriovenous intrapulmonary shunt was confirmed by reconstructed angiographic CT scan (Fig 1). A palliative left lower lobectomy led to an immediate rise in arterial oxygen saturation. At postoperative day 3, arterial blood gas values on room air showed a pH of 7.46, a PaCO
2 of 41 mm Hg; a PaO
2 of 109 mm Hg, and an oxygen saturation value of 100%. The postoperative course was uneventful, and the patient was discharged after 1 week. He was alive 6 months after the surgery.
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Fig 1. Intrapulmonary shunt associated with a bronchioloalveolar carcinoma. (A) Computed tomographic (CT) scan shows consolidation of the left lower lobe. Note enhanced pulmonary vessels and patent bronchi (angiogram sign). (B) Angio-CT scan reconstruction.
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Comment
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The first report of BAC with intrapulmonary shunting was published in 1969 [1]. We have found only nine other cases described; among which six had surgical procedures [2–6].
In previous evaluations of patients with presumed intrapulmonary shunting, the response to temporary balloon occlusion of the pulmonary artery was useful in determining those who might benefit from surgical intervention. The "CT-angiogram sign," first described by Im and colleagues [7] in 1990, may be useful instead. In BAC this may reflect an absence of the usual localized hypoxic pulmonary vasoconstriction, which seems to occur with other pulmonary conditions. The exact mechanism for this response in BAC is uncertain; perhaps this type of cancer produces angiogenic and vasodilator substances.
In these three cases, not normally considered surgical candidates, managed by surgical resection, we have demonstrated dramatic improvement in hypoxia and improvement in symptoms. Palliation has clearly been better.
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References
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- Wolinsky H, Lin A, Williams Jr MH. Lung perfusion in bronchiole-alveolar carcinoma Am Rev Resp Dis 1969;99:585-589.[Medline]
- Fishman HC, Danon J, Koopot N, Langston HT, Sharp JT. Massive intrapulmonary venoarterial shunting in alveolar cell carcinoma Amer Rev Resp Dis 1974;109:124-128.[Medline]
- Sarlin RF, Schillaci RF, Georges TN, Wilcox JR. Focal increased lung perfusion and intrapulmonary veno-arterial shunting in bronchiole-alveolar cell carcinoma Am J Med 1980;68:618-623.[Medline]
- Chetty KG, Dick C, McGovern J, Conroy RM, Mahutte CK. Refractory hypoxemia due to intrapulmonary shunting associated with bronchioloalveolar carcinoma Chest 1997;111:1120-1121.[Abstract/Free Full Text]
- Vanoyan AA, Conroy R, Dick C. Demonstration of pathologic shunting during pulmonary angiography in a case of bronchioloalveolar carcinoma J Vasc Interv Radiol 1998;9:523-524.[Medline]
- Barlesi F, Doddoli C, Thomas P, Kleisbauer JP, Giudicelli R, Fuentes P. Bilateral bronchioloalveolar carcinoma: is there a place for palliative pneumonectomy? Eur J Cardiothorac Surg 2001;20:1113-1116.[Abstract/Free Full Text]
- Im JG, Han MC, Yu EJ, et al. Lobar bronchioloalveolar carcinoma: "angiogram sign" on CT scans Radiology 1990;176:749-753.[Abstract/Free Full Text]
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Abstract
Introduction
