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Journal of Thoracic Oncology:
doi: 10.1097/JTO.0b013e3181d3ccdf
Case Reports

Resection of Mucinous Lung Adenocarcinoma Presenting with Intractable Bronchorrhea

Takao, Motoshi MD, PhD*; Takagi, Takehiro MD†; Suzuki, Hitoshi MD, PhD*; Shimamoto, Akira MD, PhD*; Murashima, Shuichi MD, PhD‡; Taguchi, Osamu MD, PhD†; Shimpo, Hideto MD, PhD*

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*Departments of General Thoracic Surgery, †Respiratory Medicine, and ‡Diagnostic Radiology, Mie University Hospital, Tsu, Mie, Japan.

Disclosure: The authors declare no conflicts of interest.

Address for correspondence: Motoshi Takao, MD, PhD, 2-174 Edobashi, Tsu, Mie 514-8507, Japan. E-mail: takao@clin.medic.mie-u.ac.jp

A 59-year-old man who was a current smoker (30 pack-years) was referred to the outpatient department of our hospital for evaluation of a huge left hilar mass with bilateral diffuse opacification of the lung fields on the chest x-ray. Although the patient had no symptoms at his first visit to our clinic, he soon developed progressive dyspnea of acute onset with hypoxemia necessitating oxygen supplementation and continuous bronchorrhea (>300 mL/d). Moreover, he could not lie on his back because of the bronchorrhea flooding out of the nose in this position. The results of arterial blood gas analysis on room air were as follows: pH, 7.422; PaO2, 60.3 torr; Paco2, 35.6 torr; HCO3, 23.2 mEq/L; BE, −0.6; O2 Sat, 91.7%. Fiberoptic bronchoscopy and transbronchial lung biopsy revealed the diagnosis of adenocarcinoma in specimens obtained from the apical segment of the left lower lobe; no malignant cells were identified in the bronchoscopic aspirates obtained from the other lobes. Although repeat sputum cytology did not reveal any malignant cells, the carcinoembryonic antigen level in the sputum was 128 mg/dL as compared with that of 4.2 mg/dL in the serum. High-resolution computed tomography (CT) revealed bilateral diffuse ground-glass attenuation (GGA) of the pulmonary parenchyma, in addition to a huge round area of consolidation in the apical segment of the left lower lobe (Figure 1). Positron emission tomography with fluorine-18 fluorodeoxyglucose showed a high standardized uptake value of 7.4 in the pulmonary tumor and regional hilar lymph nodes, with no evidence of any metastatic disease (Figure 2). We obtained informed consent from the patient for surgery after explaining to him that the surgical procedure to be undertaken may not be curative, but palliative in terms of providing relief from the disabling symptoms of bronchorrhea and dyspnea and improving the arterial oxygen saturation, and performed a left lower lobectomy with systematic radical lymph node dissection. Although a lingular segment obtained by wedge resection did not reveal any evidence of metastasis, multiple small intrapulmonary metastases were evident in the resected left lower lobe. Histopathological examination revealed adenocarcinoma with mucinous bronchioloalveolar adenocarcinoma (BAC) features (Figure 3) and hilar lymph node metastases; pT3N1M0, stage IIIA. Epidermal growth factor receptor direct sequencing of exons 18 to 22 revealed the wild type. The postoperative course was uneventful. The symptoms of bronchorrhea and dyspnea disappeared rapidly, and by the end of day 2 postoperatively, the patient no longer needed oxygen supplementation; arterial blood gas analysis at this time showed a Pao2 of 88.1 torr on room air. A postoperative CT obtained 3 weeks after the surgery (Figure 4) revealed complete disappearance of the diffuse GGA in the residual pulmonary parenchyma. Although he has taken adjuvant chemotherapy with paclitaxel and carboplatin and been well without any pulmonary symptom, a postoperative CT obtained 4 months after surgery revealed difuuse GGA in bilateral lungs suggesting intrapulmonary recurrence.

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Figure 2
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Figure 4
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DISCUSSION

Mucinous BAC and adenocarcinoma of the lung may typically present with the specific clinical features of bronchorrhea or hypoxemia caused by intrapulmonary dissemination of the disease due to bronchial or lymphatic spread. As the diffuse disease associated with the mucin-producing tumor is regarded as a metastatic disease, bilateral BAC is usually considered as a contraindication to surgery.1 Moreover, it is usually reported to be resistant to platinum-based chemotherapy and small-molecular tyrosine kinase inhibitors.2,3 Few medical strategies are available for effective palliation of the respiratory distress caused by bronchorrhea in patients with diffuse BAC, although oral erythromycin, tyrosine kinase inhibitors, or inhaled indomethacin have been reported to inhibit the bronchial hypersecretion and provide temporary relief from the symptoms in a few cases.1,4,5

We report successful treatment of a patient of mucinous lung adenocarcinoma presenting with bronchorrhea by left lower lobectomy. The surgery was performed as a palliative intent for providing relief from the disabling symptoms of bronchorrhea and dyspnea and improving the arterial oxygen saturation, rather than as curative resection. It was difficult to distinguish intrapulmonary aspiration of mucus produced in massive amounts by the tumor, manifesting as bronchorrhea, from intrapulmonary dissemination preoperatively, because GGA on high-resolution CT, the extensive alveolar filling pattern, may represent aspiration pneumonia or the “field effect” of the tumor or other modes of dissemination such as aerogenous spread and lympho-vascular permeation. The findings on the postoperative CT obtained 3 weeks after the surgery (Figure 4) revealed complete disappearance of the diffuse GGA in the residual pulmonary parenchyma might suggest that most of the diffuse GGA involving multiple lung lobes observed on the preoperative CT images was likely to have been due to bronchorrhea by the mucus producing tumor rather than to intrapulmonary metastases. As the fact that the pathology demonstrated intrapulmonary spread within the lobe worried us considerably that the bilateral changes might end up being malignant in origin despite the improvement on CT, a postoperative CT obtained 4 months after surgery revealed difuuse GGA in bilateral lungs suggesting intrapulmonary recurrence of mucinous BAC or adenocarcinoma. However, surgery could bring not only a diminution of the symptoms of dyspnea, hypoxemia, and bronchorrhea but also a chance to take systemic chemotherapy in this case. Although further follow-up is needed, surgical management of patients presenting with diffuse GGA may be considered for highly selected patients, in particular, those with the disabling symptom of intractable bronchorrhea.1

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REFERENCES

1. Garfield DH, Cadranel JL, Wislez M, et al. The bronchioloalveolar carcinoma and peripheral adenocarcinoma spectrum of disease. J Thorac Oncol 2006;1:344–359.

2. Karin EF, Lecia VS, Victoria AJ, et al. Mucinous differentiation correlates with absence of EGFR mutation and presence of KRAS mutation in lung adenocarcinomas with bronchioloalveolar features. J Mol Diagn 2007;9:320–326.

3. Vincent AM, Gregory JR, Maureen FZ, et al. Molecular characteristics of bronchioloalveolar carcinoma and adenocarcinoma, bronchioloalveolar carcinoma subtype, predict response to erlotinib. J Clin Oncol 2008;26:1472–1478.

4. Homma S, Kawabata M, Kishi K, et al. Successful treatment of refractory bronchorrhea by inhaled indomethacin in two patients with bronchioloalveolar carcinoma. Chest 1999;115:1465–1468.

5. Chetty KG, Dick C, McGovern J, et al. Refractory hypoxemia due to intrapulmonary shunting associated with bronchioloalveolar carcinoma. Chest 1997;111:1120–1121.

© 2010International Association for the Study of Lung Cancer

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