Successful Use of a Vascularized Intercostal Muscle Flap to Seal a Persistent Intrapleural Cerebrospinal Fluid Leak in a Child By Richard
G. Azizkhan,
Joseph
B. Roberson,
Jr, and Stephen
K. Powers
Chapel Hill, North Carolina l The diagnosis and management of a persistent intrapleural-dural cerebrospinal fluid fistula following excision of a large mediastinal ganglioneuroma with intraspinal extension is reported. Use of a vascularized intercostal muscle flap to close the dural fistula was curative in this I-year-old patient. Copyright o 1991 by W.B. Saunders Company INDEX WORDS: Cerebrospinal
fluid leak; dural fistula.
P
ROLONGED CEREBROSPINAL fluid (CSF) leakage following spinal trauma or surgery is unusual.’ We report the case of a 4-year-old girl who developed a persistant thoracic dural-pleural fistula with recalcitrant hydrothorax following the excision of a large posterior mediastinal ganglioneuroma with intraspinal tumor extension. In this report, we highlight the difficulties in management and describe for the first time successful surgical closure of a dural fistula using a vascularized intercostal muscle flap. CASE
REPORT
A previously healthy 4-year-old girl presented to her pediatrician with symptoms of a severe upper respiratory infection of 2 weeks’ duration. Her physical examination was normal except for some decreased breath sounds in the left upper lobe associated with some expiratory rhonchi and tactile fremitus. An &cm posterior mediastinal mass was seen on chest x-ray displacing the left upper lobe inferiorly (Fig 1). Magnetic resonance imaging demonstrated a 50% decrease in the cross-sectional area of the trachea and left mainstem bronchus from tumor compression. In addition, the mass extended into the spinal cord through the T, and T, neural foraminae and appeared as a rim of extradural tumor from T, to T,. There was no evidence of metastatic disease on bone scan and bone marrow examination. A left posterior lateral thoracotomy was used to excise the tumor. The left T, and T, pedicles were removed in order to expose the laterally placed extradural intraspinal tumor. Unfortunately, during removal of the tumor a large dural opening was encountered where the tumor had grown 2 to 3 mm intradurally along the Tz nerve root. The T3 nerve was clipped proximal to the tumor attachment at the level of the root sleeve proximal to the dorsal root ganglion; however, the T2 root had to be divided intradurally and the leaves of dura on either side of root sleeve, which was removed with the tumor, were approximated with metal vascular
From the Divisions of Pediatric Surgery and Neurosurgery, Depatiment of Pediatrics, University of North Carolina School of Medicine, Chapel Hill, NC. Address reprint requests to Richard G. A.&khan, MD, Chief Pediatric Surgery, Depamnent of Surgery, CB #7210, Universuy of North Carolina, School of Medicine, Chapel Hill, NC 27599. Copyright o 1991 by WB. Saunders Company 0022-3468/91/2606-0025$03.OOlO
744
clips. Initially, the expanded lung did not fill completely the resected tumor space, leaving a small cavity adjacent to the spinal column. A chest tube was placed for air and fluid evacuation. Except for a postoperative Horner’s syndrome, the child initially did well postoperatively and was discharged home on the 5th postoperative day. The tumor was a large ganglioneuroma by pathological evaluation. On the 14th postoperative day, the child became tachypneic. A chest x-ray demonstrated a large left hydrothorax (Fig 2). After 900 mL of fluid was initially drained through a chest tube, the child continued to drain 300 mL/d of clear but slightly xanthochromic fluid during the next 2 weeks. Cell count of the fluid did not show any lymphocytes and the measured pleural fluid triglycerides was less than serum levels. With ingestion of a heavy cream milkshake mixed with Sudan black there was no change in the color of the chest tube drainage. Repeated lumbar punctures failed to stop reaccumulation of the pleural fluid. Computed tomography myelogram demonstrated a CSF leak with extravasation of contrast into the pleural cavity at the T, to T, level (Fig 3). The chest tube was removed 14 days later because of tube dysfunction and minimal drainage. However, within 5 days the child had a recurrence of a significant loculated left hydrothorax that precipitated reoperation. One month after the original procedure, the thoracotomy was reopened and at the T, to T, level a significant CSF leak was observed coming from a 4- to 5-mm dural defect that appeared to be an extension of the original caudal torn root sleeve. An intercostal pedicle muscle flap was elevated from the 4th intercostal space at the anterior axillary line to seal this fistula with viable tissue (Fig 4). The ventral intercostal artery and vein were isolated, ligated, and divided. The periosteum was elevated from the inferior to superior rib freeing up the entire intercostal muscle with the artery. This process was extended posteriorly to the level of the vertebral bodies. In this manner an g-cm intercostal muscle flap was based on the dorsal intercostal artery and vein. The flap easily reached the area of the CSF leak, where it was sutured without tension to the periosteal and ligamentous tissue surrounding the dural fistula with 5-O prolene sutures. Two chest tubes were inserted as well as a lumbar subarachnoid drain. The CSF drainage immediately stopped and the lumbar subarachnoid drain was removed on the 3rd postoperative day. On the 5th and 6th days her chest tubes were successfully removed and the child was discharged on the 7th postoperative day with no further reaccumulation of the left pleural fluid. Twelve months postoperatively the patient has had no further problems and has a normal chest x-ray. DISCUSSION
The persistance of a thoracic dural-pleural fistula complicated by a recalcitrant CSF hydrothorax is rare and unusual, particularly when one considers that the pleural cavity has been used as an alternative site for CSF resorption in hydrocephalic patients.’ The diagnosis and management of extracranial dural fistulae may be difficult.3 When nonoperative methods fail, direct surgical approaches are warranted. A variety of operative techniques have been described, which JaurnalofPediatric Surgery, Vol26, No 6 (June), 1991: pp 744-746
INTERCOSTAL
MUSCLE
FLAP TO SEAL
A CSF LEAK
745
Fig 3. Computed tomography scan myelogram showing CSF leak at Ts foramen (arrow). Loculated hydrothorax can be seen adjacent to vertebral body. Fig 1. Preoperative posteroanterior large left posterior mediastinal tumor.
chest x-ray demonstrating
include the placement of CSF diversionary shunts (eg, lumboperitoneal or ventriculoatrial shunts); direct dural closure; and packing the site of the fistula with free muscle, fat, or fascial grafts.4 Spetzler and Wilson reported that 10 of 30 patients in their series who received surgical treatment for recurrent CSF rhinorrhea required more than one operation before the fistuia was closed.’ Others have reported faiIure rates for the closure of CSF leaks with one operation
Fig 2. Posteroanterior chest x-ray confirming tension CSF hydrothorax 2 weeks following tumor excision.
as high as 40%.6 In view of this experience, the use of a viable muscle flap to close the fistula would theoretically minimize the risk of recurrent CSF leak. The use of a vascularized intercosta1 muscle flap to close a thoracic dural fistula to our knowledge has not been previously reported. However, this type of intercostal muscle flap has been successfully applied to the repair of recurrent tracheoesophageal fistula, tracheobronchial reconstruction, and in protection of
Fig 4. Diagramatic representation of developed intercostal muscle flap (small arrow) in place sutured to dural fistula site.
746
AZIZKHAN,
a tenuous bronchial anastomosis.G8 The technique for constructing this vascularized pedical flap is simple and versatile. These flaps can be made long enough to reach any part of the posterior mediastinum and can be sutured without tension to overlay a significant
ROBERSON, AND POWERS
area if necessary. Furthermore, the patient is not left with any observable loss of function following this procedure. The use of an intercostal muscle flap may prove to be a reliable method of dural fistula closure in the thoracolumbar region for other patients.
REFERENCES 1. Harrington H, Tyler HR, Welch K: Surgical treatment of postlumbar puncture dural CSF leak causing chronic headache: Case report. J Neurosurg 57:703-707,1982 2. Ransohoff J: Ventriculopleural anastomosis in treatment of midline obstructional neoplasms. J Neurosurg 11:295-298, 1954 3. Gass H, Goldstein AS, Ruskin R, et al: Chronic postmyelogram headache, isotopic demonstration dural leak and surgical care. Arch Neurol25:168-170, 1971 4. Hubbard JL, McDonald TJ, Pearson BW, et al: Spontaneous cerebrospinal fluid rhinorrhea: Evolving concepts in diagnosis and surgical management based on the Mayo Clinic experience from 1970 through 1981. Neurosurgery 16:314-321,1985
5. Spetzler RF, Wilson CB: Management of recurrent CSF rhinorrhea of the middle and posterior fossa. J Neurosurg 49:393397,1978 6. Soriana A, Hernandez-Silverio N, Carrillo A, et al: Intercostal pedicled flap in esophageal atresia. J Pediatr Surg 22:115-l 16, 1987 7. Gustafson RA, Hrabovsky EE: Intercostal muscle and myoosseous flaps in difficult pediatric thoracic problems. J Pediatr Surg 17:541-545, 1982 8. Fell SC, Mollenkopf FP, Montefusco CM, et al: Revascularization of ischemic bronchial anastomoses by an intercostal pedicle flap. J Thorac Cardiovasc Surg 90:172-178,1985
G. Azizkhan,
Joseph
B. Roberson,
Jr, and Stephen
K. Powers
Chapel Hill, North Carolina l The diagnosis and management of a persistent intrapleural-dural cerebrospinal fluid fistula following excision of a large mediastinal ganglioneuroma with intraspinal extension is reported. Use of a vascularized intercostal muscle flap to close the dural fistula was curative in this I-year-old patient. Copyright o 1991 by W.B. Saunders Company INDEX WORDS: Cerebrospinal
fluid leak; dural fistula.
P
ROLONGED CEREBROSPINAL fluid (CSF) leakage following spinal trauma or surgery is unusual.’ We report the case of a 4-year-old girl who developed a persistant thoracic dural-pleural fistula with recalcitrant hydrothorax following the excision of a large posterior mediastinal ganglioneuroma with intraspinal tumor extension. In this report, we highlight the difficulties in management and describe for the first time successful surgical closure of a dural fistula using a vascularized intercostal muscle flap. CASE
REPORT
A previously healthy 4-year-old girl presented to her pediatrician with symptoms of a severe upper respiratory infection of 2 weeks’ duration. Her physical examination was normal except for some decreased breath sounds in the left upper lobe associated with some expiratory rhonchi and tactile fremitus. An &cm posterior mediastinal mass was seen on chest x-ray displacing the left upper lobe inferiorly (Fig 1). Magnetic resonance imaging demonstrated a 50% decrease in the cross-sectional area of the trachea and left mainstem bronchus from tumor compression. In addition, the mass extended into the spinal cord through the T, and T, neural foraminae and appeared as a rim of extradural tumor from T, to T,. There was no evidence of metastatic disease on bone scan and bone marrow examination. A left posterior lateral thoracotomy was used to excise the tumor. The left T, and T, pedicles were removed in order to expose the laterally placed extradural intraspinal tumor. Unfortunately, during removal of the tumor a large dural opening was encountered where the tumor had grown 2 to 3 mm intradurally along the Tz nerve root. The T3 nerve was clipped proximal to the tumor attachment at the level of the root sleeve proximal to the dorsal root ganglion; however, the T2 root had to be divided intradurally and the leaves of dura on either side of root sleeve, which was removed with the tumor, were approximated with metal vascular
From the Divisions of Pediatric Surgery and Neurosurgery, Depatiment of Pediatrics, University of North Carolina School of Medicine, Chapel Hill, NC. Address reprint requests to Richard G. A.&khan, MD, Chief Pediatric Surgery, Depamnent of Surgery, CB #7210, Universuy of North Carolina, School of Medicine, Chapel Hill, NC 27599. Copyright o 1991 by WB. Saunders Company 0022-3468/91/2606-0025$03.OOlO
744
clips. Initially, the expanded lung did not fill completely the resected tumor space, leaving a small cavity adjacent to the spinal column. A chest tube was placed for air and fluid evacuation. Except for a postoperative Horner’s syndrome, the child initially did well postoperatively and was discharged home on the 5th postoperative day. The tumor was a large ganglioneuroma by pathological evaluation. On the 14th postoperative day, the child became tachypneic. A chest x-ray demonstrated a large left hydrothorax (Fig 2). After 900 mL of fluid was initially drained through a chest tube, the child continued to drain 300 mL/d of clear but slightly xanthochromic fluid during the next 2 weeks. Cell count of the fluid did not show any lymphocytes and the measured pleural fluid triglycerides was less than serum levels. With ingestion of a heavy cream milkshake mixed with Sudan black there was no change in the color of the chest tube drainage. Repeated lumbar punctures failed to stop reaccumulation of the pleural fluid. Computed tomography myelogram demonstrated a CSF leak with extravasation of contrast into the pleural cavity at the T, to T, level (Fig 3). The chest tube was removed 14 days later because of tube dysfunction and minimal drainage. However, within 5 days the child had a recurrence of a significant loculated left hydrothorax that precipitated reoperation. One month after the original procedure, the thoracotomy was reopened and at the T, to T, level a significant CSF leak was observed coming from a 4- to 5-mm dural defect that appeared to be an extension of the original caudal torn root sleeve. An intercostal pedicle muscle flap was elevated from the 4th intercostal space at the anterior axillary line to seal this fistula with viable tissue (Fig 4). The ventral intercostal artery and vein were isolated, ligated, and divided. The periosteum was elevated from the inferior to superior rib freeing up the entire intercostal muscle with the artery. This process was extended posteriorly to the level of the vertebral bodies. In this manner an g-cm intercostal muscle flap was based on the dorsal intercostal artery and vein. The flap easily reached the area of the CSF leak, where it was sutured without tension to the periosteal and ligamentous tissue surrounding the dural fistula with 5-O prolene sutures. Two chest tubes were inserted as well as a lumbar subarachnoid drain. The CSF drainage immediately stopped and the lumbar subarachnoid drain was removed on the 3rd postoperative day. On the 5th and 6th days her chest tubes were successfully removed and the child was discharged on the 7th postoperative day with no further reaccumulation of the left pleural fluid. Twelve months postoperatively the patient has had no further problems and has a normal chest x-ray. DISCUSSION
The persistance of a thoracic dural-pleural fistula complicated by a recalcitrant CSF hydrothorax is rare and unusual, particularly when one considers that the pleural cavity has been used as an alternative site for CSF resorption in hydrocephalic patients.’ The diagnosis and management of extracranial dural fistulae may be difficult.3 When nonoperative methods fail, direct surgical approaches are warranted. A variety of operative techniques have been described, which JaurnalofPediatric Surgery, Vol26, No 6 (June), 1991: pp 744-746
INTERCOSTAL
MUSCLE
FLAP TO SEAL
A CSF LEAK
745
Fig 3. Computed tomography scan myelogram showing CSF leak at Ts foramen (arrow). Loculated hydrothorax can be seen adjacent to vertebral body. Fig 1. Preoperative posteroanterior large left posterior mediastinal tumor.
chest x-ray demonstrating
include the placement of CSF diversionary shunts (eg, lumboperitoneal or ventriculoatrial shunts); direct dural closure; and packing the site of the fistula with free muscle, fat, or fascial grafts.4 Spetzler and Wilson reported that 10 of 30 patients in their series who received surgical treatment for recurrent CSF rhinorrhea required more than one operation before the fistuia was closed.’ Others have reported faiIure rates for the closure of CSF leaks with one operation
Fig 2. Posteroanterior chest x-ray confirming tension CSF hydrothorax 2 weeks following tumor excision.
as high as 40%.6 In view of this experience, the use of a viable muscle flap to close the fistula would theoretically minimize the risk of recurrent CSF leak. The use of a vascularized intercosta1 muscle flap to close a thoracic dural fistula to our knowledge has not been previously reported. However, this type of intercostal muscle flap has been successfully applied to the repair of recurrent tracheoesophageal fistula, tracheobronchial reconstruction, and in protection of
Fig 4. Diagramatic representation of developed intercostal muscle flap (small arrow) in place sutured to dural fistula site.
746
AZIZKHAN,
a tenuous bronchial anastomosis.G8 The technique for constructing this vascularized pedical flap is simple and versatile. These flaps can be made long enough to reach any part of the posterior mediastinum and can be sutured without tension to overlay a significant
ROBERSON, AND POWERS
area if necessary. Furthermore, the patient is not left with any observable loss of function following this procedure. The use of an intercostal muscle flap may prove to be a reliable method of dural fistula closure in the thoracolumbar region for other patients.
REFERENCES 1. Harrington H, Tyler HR, Welch K: Surgical treatment of postlumbar puncture dural CSF leak causing chronic headache: Case report. J Neurosurg 57:703-707,1982 2. Ransohoff J: Ventriculopleural anastomosis in treatment of midline obstructional neoplasms. J Neurosurg 11:295-298, 1954 3. Gass H, Goldstein AS, Ruskin R, et al: Chronic postmyelogram headache, isotopic demonstration dural leak and surgical care. Arch Neurol25:168-170, 1971 4. Hubbard JL, McDonald TJ, Pearson BW, et al: Spontaneous cerebrospinal fluid rhinorrhea: Evolving concepts in diagnosis and surgical management based on the Mayo Clinic experience from 1970 through 1981. Neurosurgery 16:314-321,1985
5. Spetzler RF, Wilson CB: Management of recurrent CSF rhinorrhea of the middle and posterior fossa. J Neurosurg 49:393397,1978 6. Soriana A, Hernandez-Silverio N, Carrillo A, et al: Intercostal pedicled flap in esophageal atresia. J Pediatr Surg 22:115-l 16, 1987 7. Gustafson RA, Hrabovsky EE: Intercostal muscle and myoosseous flaps in difficult pediatric thoracic problems. J Pediatr Surg 17:541-545, 1982 8. Fell SC, Mollenkopf FP, Montefusco CM, et al: Revascularization of ischemic bronchial anastomoses by an intercostal pedicle flap. J Thorac Cardiovasc Surg 90:172-178,1985
