SPINE Volume 39, Number 10, pp E657-E659 ©2014, Lippincott Williams & Wilkins
CASE REPORT
Trachea-Thoracic Vertebral Body Fistula Causing Spinal Cord Injury A Case Report Tetsuya Shimokawa, MD,* Kei Miyamoto, MD, PhD,† Akira Hioki, MD, PhD,* Takahiro Masuda, MD, PhD,* Haruhiko Akiyama, MD, PhD,* and Katsuji Shimizu, MD, DMSc‡
Study Design. A case report and review of the literature. Objective. To discuss the significance of identifying tracheathoracic vertebral body fistula. Summary of Background Data. Several authors have reported the occurrence of communication between the trachea and the spinal canal or paraspinal abscess. However, trachea-thoracic vertebral body fistulas have not been reported. Methods. A 77-year-old male experienced gradually worsening lower-extremity palsy. Fifteen years before presentation, he had undergone retrosternal esophageal reconstruction with a gastric tube for esophageal cancer. Results. Computed tomographic scan revealed compression of the Th5 vertebral body; the trachea and the Th5 vertebral body were connected by a fistula, and air seemed to enter Th5 from the trachea. Magnetic resonance image showed severe spinal canal stenosis. Posterior thoracic decompression and fusion was performed. Five months after the procedure, he regained the ability to walk with a cane, and computed tomographic scan revealed closure of the fistula and the bony fusion. Conclusion. To our knowledge, this is the first case report of a trachea-thoracic vertebral body fistula. Because the trachea is adjacent to the anterior aspect of the thoracic vertebrae, in cases of retrosternal esophageal reconstruction, trachea-vertebral body fistula should be a differential diagnosis for destructive changes in thoracic vertebrae without any signs of trauma.
From the *Department of Orthopaedic Surgery, School of Medicine, Gifu University, Gifu, Japan; †Department of Reconstructive Surgery for Spine, Bone, and Joint, Gifu University Graduate School of Medicine, Gifu, Japan; and ‡Spine Center, Gifu Municipal Hospital, Gifu, Japan. Acknowledgment date: December 10, 2013. First revision date: January 21, 2014. Acceptance date: February 6, 2014. The manuscript submitted does not contain information about medical device(s)/drug(s). No funds were received in support of this work. No relevant financial activities outside the submitted work. Address correspondence and reprint requests to Tetsuya Shimokawa, MD, Department of Orthopaedic Surgery, Gifu University Graduate School of Medicine, 1-1 Yanagido, Gifu City, Gifu 501-1194, Japan; E-mail: [email protected] DOI: 10.1097/BRS.0000000000000287 Spine
Key words: trachea-thoracic vertebral body fistula, thoracic spine, trachea, spinal cord injury. Level of Evidence: N/A Spine 2014;39:E657–E659
S
everal authors have reported communication between the trachea and the spinal canal or paraspinal abscess due to trauma, surgical complications, or infection.1–5 We describe an extremely rare case of a fistula connecting the trachea to a vertebral body and causing spinal cord injury.
CASE REPORT A 77-year-old male was hospitalized for pneumonia, but he experienced gradually worsening lower-extremity palsy. A burst fracture of thoracic vertebrae 5 (Th5) was detected by computed tomography, and spinal cord compression at the same level was revealed by magnetic resonance image. Therefore, he was referred to our hospital. His medical history was significant for retrosternal esophageal reconstruction with a gastric tube for esophageal cancer 15 years ago. Manual muscle testing of the legs was reduced to 1 to 3 and urinary dysfunction was detected. Blood examination revealed no inflammatory changes. Thoracic spine computed tomographic (CT) scan at our hospital showed that the Th5 vertebral body was aerated and compressed. The trachea and the Th5 vertebral body were connected by a fistula, and air seemed to enter Th5 from the trachea (Figure 1). Magnetic resonance image showed severe spinal cord compression (Figure 2). We considered pseudarthrosis, pyrogenic spondylitis, or pathological fracture secondary to metastasis from esophageal cancer as differential diagnoses. Posterior thoracic decompression (laminectomy at Th4Th5) and fusion with instrumentation and local bone graft (Th2-Th8) were performed (Figure 3). During surgery, no air leakage or pathological fragility of the removed lamina was observed. After surgery, the patient's paralysis improved rapidly. Bronchoscopy was performed on postoperative day 6. A fistula surrounded by white fluffy granulation was detected proximal to the carina. Air passage through the fistula was www.spinejournal.com
E657
Copyright © 2014 Lippincott Williams & Wilkins. Unauthorized reproduction of this article is prohibited. SPINE131448_LR E657
01/04/14 12:12 PM
CASE REPORT
Trachea-Thoracic Vertebral Body Fistula • Shimokawa et al
Figure 1. (A) Sagittal view and (B) axial view of computed tomographic scans showing trachea-vertebral body fistula and air entering Th5 from the trachea. White arrow in A shows Th5 vertebral body, and white arrow in B shows trachea-thoracic vertebral body fistula.
confirmed by the intermittent appearance of bubbles with breathing (Figure 4). Tissue samples obtained from the fistula were negative on culture and did not show malignancy. Positron emission tomography also did not show malignancy at this site. We considered that performing anterior reconstruction using a structural graft or cage could provide better stability and radical solution for the fistula. However, respiratory surgeons’ and physicians’ team suggested that combination of latissimus dorsi muscle flap or intratracheal stenting is essential to close the fistula. They further alarmed us that such major anterior surgery for this patient with poor general condition can cause fatal complication. So, the patient was followed conservatively. Five months postoperatively, chest CT scan showed spontaneous closure of the fistula and bony fusion (Figure 5). He had been followed until postoperative 8 months when he suddenly died of lung thrombosis. Until the final period in his life, he was able to walk by himself, and
Figure 2. (A) T1WI and (B) T2WI of magnetic resonance images showing severe spinal cord compression by trachea-vertebral body fistula. White arrow in A shows Th5 vertebral body.
E658
Figure 3. After thoracic decompression and instrumented fusion from Th2 to Th8. (A) Postoperative plain radiographs anteroposterior and (B) lateral view.
there were no signs of infection. Radiographical examination showed no signs of implant loosening.
DISCUSSION To our knowledge, no studies have reported a condition resembling trachea-vertebral body fistula. A PubMed search for “spine” and “bronchial fistula” or "tracheal fistula” yielded several reports describing cases with communication between the trachea and the spine. These included cases that had air in the spinal canal associated with trauma,3 a case with spinal caries in which bronchial fistula was formed by an abscess,1 a case with spinal caries in which postoperative purulent spondylitis had developed leading to a bronchial fistula,5 and a case with anterior instrumentation of the thoracic spine in which the implant communicated with the trachea.4 The trachea is wider and less flexible than the esophagus. Therefore, if the trachea adheres to an adjacent vertebral body, the trachea may be damaged because of continuous compression and mechanical stress, resulting in a potential
Figure 4. (A) and (B) are bronchoscopy revealing a fistula proximal to the carina and the intermittent appearance of air bubbles with breathing. Black arrow in A shows fistula, and another arrow in B shows air bubble with breathing.
www.spinejournal.com
May 2014
Copyright © 2014 Lippincott Williams & Wilkins. Unauthorized reproduction of this article is prohibited. SPINE131448_LR E658
01/04/14 12:12 PM
CASE REPORT
Trachea-Thoracic Vertebral Body Fistula • Shimokawa et al
fistula. Posterior thoracic decompression and fusion resulted in spontaneous closure of the fistula. Awareness of this condition is essential for a proper differential diagnosis in rare thoracic vertebral fractures, especially in patients who have undergone a surgery for esophageal cancer.
➢ Key Points
Figure 5. (A) Sagittal and (B) axial view of computed tomographic scans showing closure of the trachea-vertebral body fistula.
opening. In this case, chest CT scan showed the retrosternal esophageal reconstruction performed for esophageal cancer 15 years ago; this reconstruction may have caused tracheal adhesion to the thoracic vertebral body. Chest CT scan obtained preoperatively showed air within the vertebral body, suggesting that the fistula was produced by a chronic morbid condition. Thoracic posterior fusion reduced the mechanical stress and enabled spontaneous closure of the fistula. Thus, the pathogenic processes of fistula formation in this case likely involved a compression fracture of Th5, which produced mechanical stress on the trachea; this condition in combination with abnormal motility (such as in pseudarthrosis) or a Th4/Th5 bone spur pressing on the trachea caused fistula formation.
CONCLUSION In summary, we report a rare case of a pathological thoracic vertebral paralytic fracture due to a trachea-vertebral body
Spine
We report a very rare case of thoracic vertebral fracture with spinal cord injury due to tracheavertebral body fistula. Posterior thoracic decompression and fusion was effective for spontaneous closure of the fistula. Awareness of this condition is essential for a proper differential diagnosis in rare thoracic vertebral fractures, especially in patients who have undergone a surgery for esophageal cancer.
References
1. Auregan G, Chakib S, Etienne B, et al. Bronchial fistula caused by Pott's abscesses. Report of 3 cases. Rev Pneumol Clin 1989;45: 175–8. 2. Derner M, Drugova B, Horejsi L, et al. Massive pneumorrhachis, pneumocephalus and pneumoopticus following thoracic trauma and avulsion of the brachial plexus: case report and review of the literature. Prague Med Rep 2011;112:56–66. 3. McIntosh SE, Salcedo-Dovi H, Cortes V. Air in the spinal canal associated with trauma. J Emerg Med 2006;31:33–5. 4. Son S, Kang DH, Choi DS, et al. A case of broncho-paraspinal fistula induced by metallic devices: delayed complication of thoracic spinal surgery. J Korean Neurosurg Soc 2011;50:64–7. 5. Masuda M, Mori H, Shiba H, et al. Pyogenic spondylitis with bronchial and skin fistula, occurred after long time post operation of tuberculous spondylitis—a case report. J West Japanese Res Soc Spine 2008;34:93–7.
www.spinejournal.com
E659
Copyright © 2014 Lippincott Williams & Wilkins. Unauthorized reproduction of this article is prohibited. SPINE131448_LR E659
01/04/14 12:12 PM
CASE REPORT
Trachea-Thoracic Vertebral Body Fistula Causing Spinal Cord Injury A Case Report Tetsuya Shimokawa, MD,* Kei Miyamoto, MD, PhD,† Akira Hioki, MD, PhD,* Takahiro Masuda, MD, PhD,* Haruhiko Akiyama, MD, PhD,* and Katsuji Shimizu, MD, DMSc‡
Study Design. A case report and review of the literature. Objective. To discuss the significance of identifying tracheathoracic vertebral body fistula. Summary of Background Data. Several authors have reported the occurrence of communication between the trachea and the spinal canal or paraspinal abscess. However, trachea-thoracic vertebral body fistulas have not been reported. Methods. A 77-year-old male experienced gradually worsening lower-extremity palsy. Fifteen years before presentation, he had undergone retrosternal esophageal reconstruction with a gastric tube for esophageal cancer. Results. Computed tomographic scan revealed compression of the Th5 vertebral body; the trachea and the Th5 vertebral body were connected by a fistula, and air seemed to enter Th5 from the trachea. Magnetic resonance image showed severe spinal canal stenosis. Posterior thoracic decompression and fusion was performed. Five months after the procedure, he regained the ability to walk with a cane, and computed tomographic scan revealed closure of the fistula and the bony fusion. Conclusion. To our knowledge, this is the first case report of a trachea-thoracic vertebral body fistula. Because the trachea is adjacent to the anterior aspect of the thoracic vertebrae, in cases of retrosternal esophageal reconstruction, trachea-vertebral body fistula should be a differential diagnosis for destructive changes in thoracic vertebrae without any signs of trauma.
From the *Department of Orthopaedic Surgery, School of Medicine, Gifu University, Gifu, Japan; †Department of Reconstructive Surgery for Spine, Bone, and Joint, Gifu University Graduate School of Medicine, Gifu, Japan; and ‡Spine Center, Gifu Municipal Hospital, Gifu, Japan. Acknowledgment date: December 10, 2013. First revision date: January 21, 2014. Acceptance date: February 6, 2014. The manuscript submitted does not contain information about medical device(s)/drug(s). No funds were received in support of this work. No relevant financial activities outside the submitted work. Address correspondence and reprint requests to Tetsuya Shimokawa, MD, Department of Orthopaedic Surgery, Gifu University Graduate School of Medicine, 1-1 Yanagido, Gifu City, Gifu 501-1194, Japan; E-mail: [email protected] DOI: 10.1097/BRS.0000000000000287 Spine
Key words: trachea-thoracic vertebral body fistula, thoracic spine, trachea, spinal cord injury. Level of Evidence: N/A Spine 2014;39:E657–E659
S
everal authors have reported communication between the trachea and the spinal canal or paraspinal abscess due to trauma, surgical complications, or infection.1–5 We describe an extremely rare case of a fistula connecting the trachea to a vertebral body and causing spinal cord injury.
CASE REPORT A 77-year-old male was hospitalized for pneumonia, but he experienced gradually worsening lower-extremity palsy. A burst fracture of thoracic vertebrae 5 (Th5) was detected by computed tomography, and spinal cord compression at the same level was revealed by magnetic resonance image. Therefore, he was referred to our hospital. His medical history was significant for retrosternal esophageal reconstruction with a gastric tube for esophageal cancer 15 years ago. Manual muscle testing of the legs was reduced to 1 to 3 and urinary dysfunction was detected. Blood examination revealed no inflammatory changes. Thoracic spine computed tomographic (CT) scan at our hospital showed that the Th5 vertebral body was aerated and compressed. The trachea and the Th5 vertebral body were connected by a fistula, and air seemed to enter Th5 from the trachea (Figure 1). Magnetic resonance image showed severe spinal cord compression (Figure 2). We considered pseudarthrosis, pyrogenic spondylitis, or pathological fracture secondary to metastasis from esophageal cancer as differential diagnoses. Posterior thoracic decompression (laminectomy at Th4Th5) and fusion with instrumentation and local bone graft (Th2-Th8) were performed (Figure 3). During surgery, no air leakage or pathological fragility of the removed lamina was observed. After surgery, the patient's paralysis improved rapidly. Bronchoscopy was performed on postoperative day 6. A fistula surrounded by white fluffy granulation was detected proximal to the carina. Air passage through the fistula was www.spinejournal.com
E657
Copyright © 2014 Lippincott Williams & Wilkins. Unauthorized reproduction of this article is prohibited. SPINE131448_LR E657
01/04/14 12:12 PM
CASE REPORT
Trachea-Thoracic Vertebral Body Fistula • Shimokawa et al
Figure 1. (A) Sagittal view and (B) axial view of computed tomographic scans showing trachea-vertebral body fistula and air entering Th5 from the trachea. White arrow in A shows Th5 vertebral body, and white arrow in B shows trachea-thoracic vertebral body fistula.
confirmed by the intermittent appearance of bubbles with breathing (Figure 4). Tissue samples obtained from the fistula were negative on culture and did not show malignancy. Positron emission tomography also did not show malignancy at this site. We considered that performing anterior reconstruction using a structural graft or cage could provide better stability and radical solution for the fistula. However, respiratory surgeons’ and physicians’ team suggested that combination of latissimus dorsi muscle flap or intratracheal stenting is essential to close the fistula. They further alarmed us that such major anterior surgery for this patient with poor general condition can cause fatal complication. So, the patient was followed conservatively. Five months postoperatively, chest CT scan showed spontaneous closure of the fistula and bony fusion (Figure 5). He had been followed until postoperative 8 months when he suddenly died of lung thrombosis. Until the final period in his life, he was able to walk by himself, and
Figure 2. (A) T1WI and (B) T2WI of magnetic resonance images showing severe spinal cord compression by trachea-vertebral body fistula. White arrow in A shows Th5 vertebral body.
E658
Figure 3. After thoracic decompression and instrumented fusion from Th2 to Th8. (A) Postoperative plain radiographs anteroposterior and (B) lateral view.
there were no signs of infection. Radiographical examination showed no signs of implant loosening.
DISCUSSION To our knowledge, no studies have reported a condition resembling trachea-vertebral body fistula. A PubMed search for “spine” and “bronchial fistula” or "tracheal fistula” yielded several reports describing cases with communication between the trachea and the spine. These included cases that had air in the spinal canal associated with trauma,3 a case with spinal caries in which bronchial fistula was formed by an abscess,1 a case with spinal caries in which postoperative purulent spondylitis had developed leading to a bronchial fistula,5 and a case with anterior instrumentation of the thoracic spine in which the implant communicated with the trachea.4 The trachea is wider and less flexible than the esophagus. Therefore, if the trachea adheres to an adjacent vertebral body, the trachea may be damaged because of continuous compression and mechanical stress, resulting in a potential
Figure 4. (A) and (B) are bronchoscopy revealing a fistula proximal to the carina and the intermittent appearance of air bubbles with breathing. Black arrow in A shows fistula, and another arrow in B shows air bubble with breathing.
www.spinejournal.com
May 2014
Copyright © 2014 Lippincott Williams & Wilkins. Unauthorized reproduction of this article is prohibited. SPINE131448_LR E658
01/04/14 12:12 PM
CASE REPORT
Trachea-Thoracic Vertebral Body Fistula • Shimokawa et al
fistula. Posterior thoracic decompression and fusion resulted in spontaneous closure of the fistula. Awareness of this condition is essential for a proper differential diagnosis in rare thoracic vertebral fractures, especially in patients who have undergone a surgery for esophageal cancer.
➢ Key Points
Figure 5. (A) Sagittal and (B) axial view of computed tomographic scans showing closure of the trachea-vertebral body fistula.
opening. In this case, chest CT scan showed the retrosternal esophageal reconstruction performed for esophageal cancer 15 years ago; this reconstruction may have caused tracheal adhesion to the thoracic vertebral body. Chest CT scan obtained preoperatively showed air within the vertebral body, suggesting that the fistula was produced by a chronic morbid condition. Thoracic posterior fusion reduced the mechanical stress and enabled spontaneous closure of the fistula. Thus, the pathogenic processes of fistula formation in this case likely involved a compression fracture of Th5, which produced mechanical stress on the trachea; this condition in combination with abnormal motility (such as in pseudarthrosis) or a Th4/Th5 bone spur pressing on the trachea caused fistula formation.
CONCLUSION In summary, we report a rare case of a pathological thoracic vertebral paralytic fracture due to a trachea-vertebral body
Spine
We report a very rare case of thoracic vertebral fracture with spinal cord injury due to tracheavertebral body fistula. Posterior thoracic decompression and fusion was effective for spontaneous closure of the fistula. Awareness of this condition is essential for a proper differential diagnosis in rare thoracic vertebral fractures, especially in patients who have undergone a surgery for esophageal cancer.
References
1. Auregan G, Chakib S, Etienne B, et al. Bronchial fistula caused by Pott's abscesses. Report of 3 cases. Rev Pneumol Clin 1989;45: 175–8. 2. Derner M, Drugova B, Horejsi L, et al. Massive pneumorrhachis, pneumocephalus and pneumoopticus following thoracic trauma and avulsion of the brachial plexus: case report and review of the literature. Prague Med Rep 2011;112:56–66. 3. McIntosh SE, Salcedo-Dovi H, Cortes V. Air in the spinal canal associated with trauma. J Emerg Med 2006;31:33–5. 4. Son S, Kang DH, Choi DS, et al. A case of broncho-paraspinal fistula induced by metallic devices: delayed complication of thoracic spinal surgery. J Korean Neurosurg Soc 2011;50:64–7. 5. Masuda M, Mori H, Shiba H, et al. Pyogenic spondylitis with bronchial and skin fistula, occurred after long time post operation of tuberculous spondylitis—a case report. J West Japanese Res Soc Spine 2008;34:93–7.
www.spinejournal.com
E659
Copyright © 2014 Lippincott Williams & Wilkins. Unauthorized reproduction of this article is prohibited. SPINE131448_LR E659
01/04/14 12:12 PM
