ENDOSCOPIC REPAIR OF ANTERIOR CRANIAL FOSSA CSF FISTULA : CASE REPORT

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1 เช ยงใหม เวชสาร 2544;40(3): Case report ENDOSCOPIC REPAIR OF ANTERIOR CRANIAL FOSSA CSF FISTULA : CASE REPORT Pongsakorn Tantilipikorn, M.D., Pichit Sittitrai, M.D. Department of Otolaryngology, Faculty of Medicine, Chiang Mai University, Chiang Mai, Thailand Abstract Endoscopic repair of an anterior cranial fossa cerebrospinal fluid (CSF) fistula has gained widespread acceptance. This study presents one case of a skull base defect after the resection of esthesioneuroblastoma. A 19-year-old man patient with a history of recurrent epistaxis was evaluated by endoscope and esthesioneuroblastoma of the right cribriform area was identified. The tumor was resected endoscopically and the skull base defect repaired by a combination of septal cartilage, temporal fascia and mucosal grafts. The septal cartilage free graft was placed above the anterior cranial fossa. This cartilage free graft served as a new framework for the anterior cranial fossa defect, which prevented a leakage of cerebrospinal fluid. The temporal fascia graft was put above the anterior cranial fossa, but beneath the septal cartilage graft in order to help mucosal migration during the healing period. After a six month postoperative period, there was neither evidence of CSF leakage nor a recurrence of the tumor. An endoscopic approach provides a safe and effective means for repairing the anterior skull base defect in selected cases. Chiang Mai Med Bull 2001;40:(3): Cerebrospinal fluid (CSF) leakage from the nostril or CSF rhinorrhea is known as a potential complication with significant morbidity and mortality. It may be either nontraumatic or traumatic in origin. Nontraumatic cases may be caused by a tumor (because of skull base erosion or high intracranial pressure), congenital anomalies, osteomyelitis of the skull and hydrocephalus. However, the traumatic cause is more common (1-2) and leaks in this condition are generally related to head injury, maxillofacial injury and iatrogenic (such as craniofacial, trans-sphenoidal and nasal surgery). The iatrogenic cause is far less likely to stop spontaneously than that following accidental trauma. (3) A craniotomy approach to repair this Address requests for reprints: Pongsakorn Tantilipikorn, M.D. Department of Otolaryngology, Faculty of Medicine, Chiang Mai University, Chiang Mai 50200, Thailand. ptantili@med.cmu.ac.th fistula has a variable success rate of 60 to 80 percents. (4-6) The inconsistent success rate and morbidity of this approach have stimulated the development of other surgical approaches. Dohlman (7) first described an extracranial approach and used a naso-orbital incision to repair an anterior cranial fossa CSF leak. Wigand (8) was the first to report on the use of endoscopes in the repair of small CSF leaks that occured during sinuse surgery. Mattox (9) also published the series of endoscopic repair. His success rate was 85.7% after the initial procedure and 100% after a second one. In this paper, the authors review the experience of endoscopic repair to skull base defects associated with CSF rhinorrhea. Case presentation A 19 year-old-man presented nasal bleeding for 30 minutes at the ENT clinic. He had a history of nasal bleeding twice

2 156 Pongsakorn T and Sittitrai P. เช ยงใหม เวชสาร prior to this presentation. On those two occasions, the examination of the anterior nasal cavities revealed no abnormality after the bleeding had been stopped by anterior nasal packing,. On this third occasion, a 0 o rigid endoscope was used after the anterior nasal packing had been removed to evaluate his nasal cavities, which revealed a bright red mass at the right cribriform area. There was no abnormal lesion in the left nasal cavity or nasopharynx. Provisional diagnosis of an olfactory nerve tumor was made and a biopsy was carried out under a rigid endoscope. There was scant bleeding after punch biopsies and no packing was needed. A pathology resulted in esthesioneuroblastoma. A computerized tomography scan revealed the tumor mass at the right cribriform area, with no evidence of brain tissue invasion, but there was a sign of anterior skull base erosion. The treatment planned was a combination of surgery and radiation. A neurosurgeon was consulted regarding the possibility of an anterior skull base defect after resection. The first plan was to repair under endoscopy, with a back-up of repairing the skull base defect by craniotomy. Operative technique The surgical procedure was carried out under general anesthesia in a supine position. The nasal cavities were packed by topical vasoconstrictor. One percent of Xylocaine with 1:50,000 epinephrine was injected inside the nose to effect the purpose of the vasoconstrictor. The tumor mass was identified at the right cribriform area, which extended down to the level of the right middle turbinate and from the anterior end to the middle part of the right middle turbinate. An infundibulotomy was completed and the ethmoid bulla showed no tumor extension. An endoscopic anterior ethmoidectomy was also performed. A ground lamella of the middle turbinate (basal lamella) was perforated and a posterior ethmoidotomy was carried out. After the skull base of the posterior ethmoid was identified, a complete anterior ethmoidectomy was performed until the skull base of the anterior ethmoid sinus was seen. There was also no extension of the tumor mass. The right middle turbinate was resected for accessibility to the tumor and as a source for the free mucosal graft. The tumor was removed from the skull base by ethmoid forceps. The skull base defect (size 2 X 1 cm) was identified with no evidence of brain tissue invasion. During tumor removal, the cerebrospinal fluid leaked continuously through the skull base defect. A septal cartilage was harvested as a free graft and a mucosal incision was performed on the contralateral side. The septal cartilage (size 3x2 cm) which included perichondrium on one side, was harvested. There was no septal mucosal perforation on the right side of the nasal cavity. A temporal fascia graft (size 3x3 cm) was also taken as a free graft. The mucosa around the skull base defect was de-epithelized. The septal cartilage was inserted and adjusted through the skull base defect until lying above the anterior cranial fossa. No cerebrospinal fluid leak was identified at this time. The temporal fascia graft was put above the anterior cranial fossa, but beneath the septal cartilage graft for the purpose of a bridge for mucosal migration during the healing period. A free mucosal graft from the resected middle turbinate was placed on the undersurface of the temporal fascia graft and secured in place by gelfoam. The anterior nasal packing was carried out with gauze and kept in place for one week. The patient was instructed to refrain from nose blowing, lifting heavy material or straining. Result The patient was discharged from hospital on the 7 th postoperative day without evidence of a CSF leak. He was scheduled for

3 ป ท 40 ฉบ บท 3 ก นยายน 2544 Endoscopic repair of CSF fistula 157 radiation, which started from the 14 th day postoperative. After radiation for one month, he had symptoms of rhinitis. From the endoscopic finding, his nasal mucosa was inflamed and the skull base showed a minimal leak of CSF. There was also crust and mucoid dischage in both sides of the nasal cavity. He was hospitalized and received an intravenous antibiotic (Cefuroxime acetyl) for two weeks. The CSF leakage ceased and the patient continued radiotherapy. All of the postoperative visits were made by an otolaryngologist. The patient s nose was examined by endoscope and photographed at the skull base defect area. For a six month postoperative period, neither a CSF leak nor recurrence of the tumor was detected. Discussion CSF rhinorrhea traditionally posed a difficult problem. The potential hazards included meningitis and pneumocephalus. The etiologies of the leak included iatrogenic, accidental, tumor and hydrocephalus. This study presented the case of an olfactory tumor that was resected and the skull base defect repaired endoscopically. Esthesioneuroblastoma is a rare tumor arising from the olfactory epithelium. Patients tend to present with an advanced stage of the disease because of the nonspecific presenting signs and symptoms. The patient in this study also presented with recurrent symptom of epistaxis, which formed the opinion of performing endoscopic examination. Early detection of the small sized tumor provided an alternative surgical procedure to the craniofacial resection, which is considered a conventional surgical treatment in this case. (10) In most cases, the treatment consists of combined-modality therapy, including radiotherapy and surgery for the early stage, and the addition of chemotherapy for late the stage. (11-13) Richtsmeier et al. (14) concluded that craniofacial resection was a relatively safe, versatile, and effective procedure for surgical management of the tumors involving the anterior skull base. Walch et al. (15) and Stammberger et al. (16) proposed that minimally invasive therapy and endoscopic sinus surgery provided a reliable new approach for the treatment of olfactory neuroblastoma. This offered an excellent quality of life with less injury to the patient, fewer side-effects, and fewer long-term effects than other treatment strategies. In this case, with skull base and dural invasion, the endoscopic approach suited very well with the probability of the repairing skull base. The conventional approaches for repairing to anterior skull base defect are the extracranial route (naso-orbital incision) (17) or craniotomy with pericranial flap. (14) These approaches to repair the defect of the anterior skull base have a variable success rate of 60 to 80 percents. (4-6) The morbidity associated with craniotomy such as a loss in the ability to smell and prolonged hospitalization has made the endoscopic approach an alternative option. In the series of endoscopic repair, the success rate was 85.7% after the initial procedure and 100% after a second one. (9) The endoscopic approach should be considered as the preferred method if the skull base defect is endoscopically accessible. Lanza et al. (18) recommended endoscopic repair for CSF fistula from various causes such as trauma, encephalocele and post tumor resection. The clear anatomical exposure of the roof of the nasal and sinus cavities by the endoscope offered the surgeon an opportunity to identify the area of the CSF leak, in which enabled an adequatel plan for treatment. (19) The application of endoscopic techniques to the closure of CSF leak had a number of advantages over conventional methods, including: (9) 1. Excellent field of vision, allowing exact localization of the leak. 2. The ability to clean mucosa from the bony defect precisely.

4 158 Pongsakorn T and Sittitrai P. เช ยงใหม เวชสาร Fig. 1. Coronal CT scan shown tumor (white arrow) at right cribiform area Fig.2. Endoscopic view shows right middle tubinate (back arrow) and tumor at right cribriform area (white arrow)

5 ป ท 40 ฉบ บท 3 ก นยายน 2544 Endoscopic repair of CSF fistula 159 Fig.3. Endoscopic view shows the edge of skull base defect (back arrow) and septal cartilagegraft (white arrow) being adjusted to cover skull defect. 3. Accurate position of the graft material over the defect. In Thailand, Jareoncharsri et al. (20) presented one case of spontaneous CSF located at the sphenoid sinus, which was repaired by a septal flap and fibrin glue. There are a variety of options for techniques to repair the defect of the anterior skull base, but the principle concept is still the same: water-tight closure. The mucoperiosteal flaps from various donor sites (especially the nasal septal) was rotated to the leak area to seal the defect, with a success rate of %. (21-22) The disadvantage of usinga pedicle flap is the possibility of its excessive tension. This may result in further retraction and subsequent loss of the graft or flap with recurrent CSF rhinorrhea. The size of defect is another factor that needs to be considered. Marks (23) advised that a small sized defect did not need anything more than a mucosal graft or flap. However, in a larger defect, a bone or cartilage graft was an essential part in the repairing process. (23) In the case of this study, we chose the septal cartilage was chosen as a free graft because of its property of pliability, which made it easier to insert through the skull base defect. Utilizing the temporal fascia in this reported case was in order to promote mucosalization. After inserting the septal cartilage and temporal fascia, the free mucosal graft was placed to cover the cartilage and the fascia in the nasal cavity side of the skull base. The mucosal graft was secured in place by gelfoam. Some studies advocated fibrin glue as an adjunction. In the study of Gassner et al, the use of fibrin glue to fix a free graft did not improve the result. (24) The problem of post-operative radiation mucositis might deteriorate the success rate. The patient in this study was treated by intravenous antibiotics and hospitalized for bed rest. The leakage rate decreased and stopped after two weeks of treatment. The

6 160 Pongsakorn T and Sittitrai P. เช ยงใหม เวชสาร radiation effect on neovascularization was possibly the reason for the recurrent CSF leak post-operatively. The patient remained disease-free with no evidence of leakage after six months. Nevertheless, close-observation is still mandatory. The average time span before recurrent esthesioneuroblastoma can be detected is more than 6 years, which is far longer than that expected for other sinonasal malignancies. (25) This study agreed that craniofacial resection with post-operative radiation remains the standard treatment in most esthesioneuroblastoma cases. With the size and location of the tumor in the patient in this study, the endoscopic approach was suitable as an alternative option. Closing of the skull base defect endoscopically has many advantages over the craniotomy approach, and the craniotomy approach is still the back-up procedure if the endoscopic one fails. Conclusion The endoscope is a valuable tool in the evaluation and subsequent treatment of selected cases of sinonasal malignancies and anterior skull base defect. Where the skull base defect is accessible by the endoscope, this study proposes that an endoscopic approach should be considered as the firstline of management. References 1. Cairns H. Injuries of the frontal and ethmoid sinuses with special references to cerebrospinal rhinorhea and aeroceles. J Layngol Otol 1937;52: Omaya AK, Di Chiro G, Baldwin M, Pennybacker JB. Nontraumatic cerebrospinal fluid rhinorrhea. J Neurol Neurosurg Psychiatry 1968;31: Carmel PW, Komisar A. Cerebrospinal fluid rhinorrhea. In: Blitzer A, Lawson W, Friedman WH, editors. Surgery of the paranasal sinuses. 2 nd edition. Philadelphia: W.B.Saunders, p Park JI, Strelzow VV, Friedman WH. Current management of cerebrospinal fluid rhinorrhea. Laryngosope 1983;93: Aarabi B, Leibrock LG. Neurosurgical approaches to cerebrospinal fluid rhinorrhea. Ear Nose Throat J 1992;71: Hubbard JL, McDonald TC, Pearson BW, et al. Spontaneous cerebrospinal fluid rhinorrhea: evolving concepts in diagnosis and management based on the Mayo Clinic experience from 1970 to 1981 Neurosurgery 1985;16: Dohlman G. Spontaneous cerebrospinal rhinorrhea. Acta Otolaryngol Suppl (Stockh) 1948;67: Wigand WE. Transnasal ethmoidectomy under endoscopic control. Rhinology 1981;19: Mattox DE, Kennedy DW. Endoscopic management of cerebrospinal fluid leaks and encephaloceles. Laryngoscope 1990;100: DeSanto LW. Neoplasms. In: Cummings CW, editor. Otolaryngology-Head and Neck Surgery. 2 nd edition. St. Louis: Mosby-Year Book, p Levine PA, Debo RF, Meredithh SD, Jane JA, Constable WC, Cantrell RW. Craniofacial resection at the University of Virginia ( ): survival analysis. Head Neck 1994;16: Brunet J, Sola C, Mesia R, et al. Esthesioneuroblastoma: experience from 4 cases treated with radiotherapy and neoadjuvant chemotherapy. Acta Otorhinolaringol Esp 1995; 46: Dulguerov P, Calcaterra T. Esthesioneuroblastoma: the UCLA experience Laryngoscope 1992;102: Richtsmeier WJ, Briggs RJ, Koch WM, et al. Complications and early outcome of anterior craniofacial resection. Arch Otolaryngol Head Neck Surg 1992;118: Walch C, Stammberger H, Anderhuber W, Unger F, Kole W, Feichtinger K. The minimally invasive approach to olfactory neuroblastoma: combined endoscopic and stereotactic treatment. Laryngoscope 2000;110: Stammberger H, Anderhuber W, Walch C, Papaefthymiou G. Possibility and limitations of endoscopic management of nasal and paranasal sinus malignancies. Acta Otorhinolaryngol Belg 1999;53: Applebaum EL, Chow JM. CSF Leaks. In: Cummings CW, editor. Otolaryngology-Head and Neck Surgery. 2 nd edition. St.Louis: Mosby-Year Book; p Lanza DC, O Brien DA, Kennedy DW. Endoscopic repair of cerebrospinal fluid fistulae and encephaloceles. Laryngoscope 1996;106:

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