JSTR MEETING NOTES
Bronchial Stump Aspergillosis Daisuke Maki, MD, PhD,* Yoshimi Mitogawa, MD,* Ayuko Yuzurio, MD,* Atushi Yoshida, MD, PhD,* and Susumu Kanazawa, MD, PhDw
Key Words: Aspergillus, postoperative infection, positron emission tomography/computed tomography
(J Thorac Imaging 2013;28:W121–W122)
A
spergillus infection limited to the tracheobronchial mucosa has been described with pseudomembranous necrotizing tracheobronchial aspergillosis in immunosuppressed patients.1 Bronchial stump aspergillosis (BSA) is a rare form of Aspergillus infection arising from the inflammatory tissue surrounding the endobronchial suture after pulmonary resection and does not relate to the patient’s immune status.2 Symptoms, including cough and hemoptysis, usually appear months after surgery.2 We report a case of a woman with BSA after a left upper lobectomy that caused severe stenosis of the left main bronchus.
FIGURE 1. Contrast-enhanced chest CT on admission revealing infiltrating soft tissue in the left hilum of the lung with stenosis of the left main bronchus.
DISCUSSION CASE REPORT A 70-year-old woman presented at our hospital with a 1-month history of severe cough. She had undergone a left upper lobectomy for bronchogenic squamous cell carcinoma 20 years ago. She was a never-smoker and had no other significant medical history. The physical examination findings were normal, except for a reduction of left breath sounds. Her laboratory data on admission (including the tumor markers carcinoembryonic antigen, progastrin-releasing peptide, neuron-specific enolase, and squamous cell carcinoma antigen) were normal. Chest computed tomography (CT) on admission (Fig. 1) revealed severe stenosis of the left main bronchus involving the soft tissue along the left main bronchus and extending into the mediastinum. Positron emission tomography/CT (PET/CT) (Fig. 2) demonstrated increased fluorodeoxyglucose activity in the soft tissue [maximum standardized uptake value (SUVmax) = 7.7]. Severe stenosis of the left main bronchus was identified upon bronchoscopy; however, there was no remarkable lesion of the bronchial mucosa. An endobronchial biopsy using forceps revealed a silk thread at the bronchial stump. Pathologic examination (Fig. 3) revealed numerous hyphae of Aspergillus species. Because the patient’s symptoms did not improve 1 month after removal of the suturing thread from the bronchial stump, itraconazole was administered orally. Chest CT 8 months after administration of the itraconazole (Fig. 4) demonstrated a reduction of the soft tissue, but the stenosis of the left main bronchus remained.
From the *Department of Radiology, Kure Kyosai Hospital, Hiroshima; and wDepartment of Radiology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, Okayama, Japan. The authors declare no conflicts of interest. Reprints: Daisuke Maki, MD, PhD, Department of Radiology, Kure Kyosai Hospital, 2-3-28 Nishityuo, Kure, Hiroshima 737-8505, Japan (e-mail: [email protected]). Copyright r 2013 by Lippincott Williams & Wilkins
J Thorac Imaging
Volume 28, Number 6, November 2013
In 1969, Sawasaki et al2 reported that the use of silk thread sutures in pulmonary resection played a principal role in causing the development of BSA in 9 patients. The incidence of BSA with silk sutures was about 1.5%, whereas no complication was encountered in 140 resections that used nylon monofilament sutures.2 In addition to an inflammatory reaction to the silk thread as a foreign body, the capillarity and gluten of the silk may accelerate the development of BSA.2 The period from the operation to the onset of BSA ranged from 6 to 12 months, with the exception of 2 cases (26 and 38 mo).2 Other materials have replaced silk sutures in surgery; however, BSA still warrants attention because some cases with onset several years after the operation have been reported.2,3 Our patient exhibited soft tissue infiltrating the left hilum of the lung with stenosis of the left main bronchus, resembling fibrosing mediastinitis. Chronic fibrosing mediastinitis due to Aspergillus has been previously
FIGURE 2. PET/CT scan showing increased fluorodeoxyglucose activity in the soft tissue (SUVmax = 7.7).
www.thoracicimaging.com |
W121
Maki et al
FIGURE 3. Numerous hyphae of Aspergillus species.
reported in an immunocompetent 78-year-old woman.4 Mediastinal aspergillosis may result from a primary Aspergillus pneumonia that spreads to the adjacent mediastinal structures, usually in an immunocompromised host. Fibrosing mediastinitis is a disorder characterized by an excessive fibrotic reaction in the mediastinum. Although many cases are idiopathic, most cases in the United States are thought to be caused by an abnormal immunologic response to Histoplasma capsulatum infection.4 PET/CT is a useful tool for detecting biochemically active lesions especially in the diagnosis, staging, and evaluation of therapy in malignancy. However, many positive findings have been reported in infectious or noninfectious inflammatory diseases and benign tumors. A similar case in which BSA was associated with increased fluorodeoxyglucose activity on a PET/CT has been reported.5 The diagnosis of BSA should be considered when follow-up PET/CT scans on bronchial stumps after lung resection are positive. Removal of the silk sutures is generally the treatment of choice for BSA.2,3 The use of oral itraconazole, endoscopic suture scissors, or Nd:YAG laser may represent valid alternatives, if removal of the sutures is not feasible.3 In the case that we describe in this report, oral itraconazole was effective, although the stenosis of the left main bronchus remained, likely due to fibrotic change.
W122 | www.thoracicimaging.com
J Thorac Imaging
Volume 28, Number 6, November 2013
FIGURE 4. Chest CT 8 months after administration of oral itraconazole showing a reduction of the soft tissue, but the stenosis of the left main bronchus remained.
In conclusion, although BSA has only been rarely observed in recent years, the possibility of BSA should still be considered in the differential diagnosis of a local recurrence. BSA was observed in the case described in this report, which occurred 20 years postoperatively.
REFERENCES 1. Huang HD, Li Q, Huang Y, et al. Pseudomembranous necrotizing tracheobronchial aspergillosis: an analysis of 16 cases. Chin Med J. 2012;125:1236–1241. 2. Sawasaki H, Horie K, Yamada M, et al. Bronchial stump aspergillosis experimental and clinical study. J Thorac Cardiovasc Surg. 1969;58:198–208. 3. Noppen M, Claes I, Maillet B, et al. Three cases of bronchial stump aspergillosis: unusual clinical presentations and beneficial effect of oral itraconazole. Eur Respir J. 1995;8:477–480. 4. Wightman SC, Kim AW, Proia LA, et al. An unusual case of Aspergillus fibrosing mediastinitis. Ann Thorac Surg. 2009;88: 1352–1354. 5. Garcia-Olive´ I, Andreo F, Rosin˜ol O, et al. Bronchial stump aspergillosis after lobectomy for lung cancer as an unusual cause of false positive fluorodeoxyglucose positron emission tomography and computed tomography: a case report. J Med Case Rep. 2011;5:72.
r
2013 Lippincott Williams & Wilkins
Bronchial Stump Aspergillosis Daisuke Maki, MD, PhD,* Yoshimi Mitogawa, MD,* Ayuko Yuzurio, MD,* Atushi Yoshida, MD, PhD,* and Susumu Kanazawa, MD, PhDw
Key Words: Aspergillus, postoperative infection, positron emission tomography/computed tomography
(J Thorac Imaging 2013;28:W121–W122)
A
spergillus infection limited to the tracheobronchial mucosa has been described with pseudomembranous necrotizing tracheobronchial aspergillosis in immunosuppressed patients.1 Bronchial stump aspergillosis (BSA) is a rare form of Aspergillus infection arising from the inflammatory tissue surrounding the endobronchial suture after pulmonary resection and does not relate to the patient’s immune status.2 Symptoms, including cough and hemoptysis, usually appear months after surgery.2 We report a case of a woman with BSA after a left upper lobectomy that caused severe stenosis of the left main bronchus.
FIGURE 1. Contrast-enhanced chest CT on admission revealing infiltrating soft tissue in the left hilum of the lung with stenosis of the left main bronchus.
DISCUSSION CASE REPORT A 70-year-old woman presented at our hospital with a 1-month history of severe cough. She had undergone a left upper lobectomy for bronchogenic squamous cell carcinoma 20 years ago. She was a never-smoker and had no other significant medical history. The physical examination findings were normal, except for a reduction of left breath sounds. Her laboratory data on admission (including the tumor markers carcinoembryonic antigen, progastrin-releasing peptide, neuron-specific enolase, and squamous cell carcinoma antigen) were normal. Chest computed tomography (CT) on admission (Fig. 1) revealed severe stenosis of the left main bronchus involving the soft tissue along the left main bronchus and extending into the mediastinum. Positron emission tomography/CT (PET/CT) (Fig. 2) demonstrated increased fluorodeoxyglucose activity in the soft tissue [maximum standardized uptake value (SUVmax) = 7.7]. Severe stenosis of the left main bronchus was identified upon bronchoscopy; however, there was no remarkable lesion of the bronchial mucosa. An endobronchial biopsy using forceps revealed a silk thread at the bronchial stump. Pathologic examination (Fig. 3) revealed numerous hyphae of Aspergillus species. Because the patient’s symptoms did not improve 1 month after removal of the suturing thread from the bronchial stump, itraconazole was administered orally. Chest CT 8 months after administration of the itraconazole (Fig. 4) demonstrated a reduction of the soft tissue, but the stenosis of the left main bronchus remained.
From the *Department of Radiology, Kure Kyosai Hospital, Hiroshima; and wDepartment of Radiology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, Okayama, Japan. The authors declare no conflicts of interest. Reprints: Daisuke Maki, MD, PhD, Department of Radiology, Kure Kyosai Hospital, 2-3-28 Nishityuo, Kure, Hiroshima 737-8505, Japan (e-mail: [email protected]). Copyright r 2013 by Lippincott Williams & Wilkins
J Thorac Imaging
Volume 28, Number 6, November 2013
In 1969, Sawasaki et al2 reported that the use of silk thread sutures in pulmonary resection played a principal role in causing the development of BSA in 9 patients. The incidence of BSA with silk sutures was about 1.5%, whereas no complication was encountered in 140 resections that used nylon monofilament sutures.2 In addition to an inflammatory reaction to the silk thread as a foreign body, the capillarity and gluten of the silk may accelerate the development of BSA.2 The period from the operation to the onset of BSA ranged from 6 to 12 months, with the exception of 2 cases (26 and 38 mo).2 Other materials have replaced silk sutures in surgery; however, BSA still warrants attention because some cases with onset several years after the operation have been reported.2,3 Our patient exhibited soft tissue infiltrating the left hilum of the lung with stenosis of the left main bronchus, resembling fibrosing mediastinitis. Chronic fibrosing mediastinitis due to Aspergillus has been previously
FIGURE 2. PET/CT scan showing increased fluorodeoxyglucose activity in the soft tissue (SUVmax = 7.7).
www.thoracicimaging.com |
W121
Maki et al
FIGURE 3. Numerous hyphae of Aspergillus species.
reported in an immunocompetent 78-year-old woman.4 Mediastinal aspergillosis may result from a primary Aspergillus pneumonia that spreads to the adjacent mediastinal structures, usually in an immunocompromised host. Fibrosing mediastinitis is a disorder characterized by an excessive fibrotic reaction in the mediastinum. Although many cases are idiopathic, most cases in the United States are thought to be caused by an abnormal immunologic response to Histoplasma capsulatum infection.4 PET/CT is a useful tool for detecting biochemically active lesions especially in the diagnosis, staging, and evaluation of therapy in malignancy. However, many positive findings have been reported in infectious or noninfectious inflammatory diseases and benign tumors. A similar case in which BSA was associated with increased fluorodeoxyglucose activity on a PET/CT has been reported.5 The diagnosis of BSA should be considered when follow-up PET/CT scans on bronchial stumps after lung resection are positive. Removal of the silk sutures is generally the treatment of choice for BSA.2,3 The use of oral itraconazole, endoscopic suture scissors, or Nd:YAG laser may represent valid alternatives, if removal of the sutures is not feasible.3 In the case that we describe in this report, oral itraconazole was effective, although the stenosis of the left main bronchus remained, likely due to fibrotic change.
W122 | www.thoracicimaging.com
J Thorac Imaging
Volume 28, Number 6, November 2013
FIGURE 4. Chest CT 8 months after administration of oral itraconazole showing a reduction of the soft tissue, but the stenosis of the left main bronchus remained.
In conclusion, although BSA has only been rarely observed in recent years, the possibility of BSA should still be considered in the differential diagnosis of a local recurrence. BSA was observed in the case described in this report, which occurred 20 years postoperatively.
REFERENCES 1. Huang HD, Li Q, Huang Y, et al. Pseudomembranous necrotizing tracheobronchial aspergillosis: an analysis of 16 cases. Chin Med J. 2012;125:1236–1241. 2. Sawasaki H, Horie K, Yamada M, et al. Bronchial stump aspergillosis experimental and clinical study. J Thorac Cardiovasc Surg. 1969;58:198–208. 3. Noppen M, Claes I, Maillet B, et al. Three cases of bronchial stump aspergillosis: unusual clinical presentations and beneficial effect of oral itraconazole. Eur Respir J. 1995;8:477–480. 4. Wightman SC, Kim AW, Proia LA, et al. An unusual case of Aspergillus fibrosing mediastinitis. Ann Thorac Surg. 2009;88: 1352–1354. 5. Garcia-Olive´ I, Andreo F, Rosin˜ol O, et al. Bronchial stump aspergillosis after lobectomy for lung cancer as an unusual cause of false positive fluorodeoxyglucose positron emission tomography and computed tomography: a case report. J Med Case Rep. 2011;5:72.
r
2013 Lippincott Williams & Wilkins
