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Solitary Fibrous Tumour of the sinonasal cavity � Case report of endoscopic resection using coblation

Authors:
R Edmiston MBBS(Hons)1	Core Trainee
P Pal MBBS MDPath FRCPath2	Consultant NeuroPathologist
K Gnanalingham PhD FRCS SN3,4	Consultant Neurosurgeon
RK Bhalla BSc(Hons) FRCS MD1,4	Consultant Rhinologist and Endoscopic Skull Base Surgeon

1Tameside NHS Foundation Trust, Ashton-under-Lyne, UK.
2Departments of Neuropathology and Neurosurgery Greater Manchester Neurosciences Centre, 3Salford Royal Foundation Trust, Manchester, UK.
4Manchester Academic Health Sciences Centre, University of Manchester, Manchester, UK.

Running title: Solitary Fibrous tumour
Key words: Solitary Fibrous tumour; Anterior skull base; Endoscopy; Coblation


Address for correspondence:
Mr RK Bhalla
Department of Otolaryngology - Head & Neck Surgery
Manchester Royal Infirmary	
Oxford Road	
Manchester M13 9WL	
Tel: (0161) 276 � 4426
Fax: (0161) 276 � 5003
Email:   HYPERLINK "mailto:rajiv.bhalla@cmft.nhs.uk" rajiv.bhalla@cmft.nhs.uk

 
ABSTRACT 
Background:
Solitary fibrous tumours (SFT) are extremely rare mesenchymal tumours. Surgery can be complicated by bleeding. This case describes the treatment by endoscopic approach, using novel operative strategies in a high-risk patient who was unable to have blood transfusion.

Case report:
This 28 year old Caucasian female underwent trans-nasal surgery 8 years previously for a right sided sinonasal SFT. She re-presented on this occasion with recurrence of symptoms. Imaging revealed a part calcified mass at the base of anterior skull base with involvement of the right cribriform plate and with intra-cranial extension. Peri-operative embolization was found to be too hazardous.

Conclusions:
To minimise surgical trauma, a trans-nasal endoscopic approach was undertaken, and macroscopic clearance was achieved primarily using coblation. This novel technique, which is as yet unreported in the literature for sinonasal SFT, was found to be a very effective means of ensuring haemostasis whilst maintaining an excellent field of view. 

 
Key words: Solitary Fibrous tumour; Anterior skull base; Endoscopy; Coblation
INTRODUCTION: 
Solitary fibrous tumours (SFT) are extremely rare mesenchymal tumours with fewer than 200 head and neck cases reported in the literature. Although generally benign, malignancy is a risk.  They present typically with compressive symptoms and can be difficult to distinguish from other tumours of soft tissue origin.  Surgical excision is the primary mode of treatment.  The majority of patients with recurrent disease are due to incomplete resection margins following primary surgery. 

The resection of such cases can be complicated by difficulty in anatomical access and the vascular nature of the tumour. Significant intra-operative bleeding is also a concern.  Peri-operative embolisation of feeding vessels can be an useful adjunct, particularly when working in the confined space of the sinonasal vault. Coblation is a novel technique, which has been demonstrated to provide good levels of haemostasis in similar tumours of vascular origin.  However, its use on SFTs is yet to be demonstrated.

The present case combines different minimal access surgical techniques to treat a recurrent anterior skull base SFT in a patient who was unable to have blood transfusion.  





CASE REPORT:
This 28 year old Caucasian female originally presented to ENT services, elsewhere 8 years previously with a history of nasal obstruction.  She was otherwise fit and well. For religious reasons she was unwilling to have any blood products during surgery. 

Initial endoscopic examination identified a right-sided unilateral large nasal polyp and the nasal septum was pushed over to the left. Imaging performed at that time confirmed no intracranial involvement, and biopsy was reported to show a transitional papilloma.  She was readmitted for endoscopic surgical excision and septoplasty, performed by an experienced FESS surgeon. Intra-operatively it was discovered that the mass was arising between the origin of the right middle turbinate and the cribriform plate and attempts to debulk the mass resulted in significant bleeding.  As a result only a partial resection was feasible. 

The patient remained well with no clinical or radiological concerns until 8 years later when she developed symptoms of recurrent nasal obstruction and blood-stained nasal discharge.  Repeat imaging by CT and MR revealed a recurrent and largely calcified bony lesion predominantly within the right nasal cavity involving the ethmoids and extending from the back of the frontal sinus to just in front of the sphenoid sinus in the sagittal plane (Figure 1a-c).  The lesion extended between the medial walls of the orbits bilaterally with evidence of invasion through the cribriform plate with some indentation of the frontal lobes (Figure 1).  Radiologically the lesion had increased in size.  

Clinical examination at this stage was unremarkable, with intact cranial nerves and a full range of eye movements. Repeat endoscopic evaluation revealed a fleshy mass arising from the medial aspect of the right middle turbinate.

The patient�s case was discussed at the local skull base multi-disciplinary team meeting and the consensus was to proceed with transnasal surgery.  A preoperative angiogram revealed limited tumour blush, mostly arising from the right anterior ethmoidal artery. Embolisation of the latter, via the right ophthalmic artery was deemed too hazardous given the small risk of occlusion of the parent vessel with risk for blindness in that eye. So alternative techniques were sought.  

An extended transnasal approach was undertaken, with the use of image guidance (Brainlab, Munich, Germany), microdebrider (Medtronic, Minneapolis, USA) and coblator (Procise EZ View � settings 9-3, Arthrocare, Austin, Texas). Through a binostril approach, a Hadad-Bassagasteguy nasoseptal flap was prepared. After bilateral middle turbinectomies the firm, vascular and partly calcified tumour arising from the base of the right middle turbinate was encountered. The lesion was resected in piecemeal fashion, primarily using the coblator on the solid non-calcified component of the tumour. A high speed diamond drill and the microdebrider was used to resect the more calcified central component of the tumour and towards the end, the blood supply from the right anterior ethmoidal artery was divided by diathermy. That part of the tumour eroding through the right cribriform plate was resected en-bloc with the involved dura. Estimated total blood loss was less than 500mls.

The large anterior skull base defect was reconstructed with haemostatic gelatin sponge (Spongostan, Ethicon, UK), covered in a multi-layered fashion by a fascia lata graft (from right thigh incision), the nasoseptal flap and a hydrogel sealant (Duraseal, Confluent Surgical, US). This was further supported by self reabsorbable Nasopore dressing (Polyganics, Groningen,( The Netherlands) and bilateral nasal packs (Merocel, Medtronic, USA). The nasal packs were removed 24hrs later and the patient was also placed on a 10 day course of prophylactic antibiotics (Augmentin; 625mg tds oral), according to local protocols.

There were no early post-operative complications including CSF leak or meningitis.  At early follow-up, the patient reported nasal crusting which gradually subsided over a 2-3 month period with daily saline nasal spray and monthly endoscopic debridement as an outpatient. Endoscopic examination at 6-months after surgery revealed complete re-epithelialisation of the anterior skull base mucosa. As expected, following such a procedure she is hypoosmic, but her sense of taste was normal. Follow-up MR confirmed macroscopic clearance of tumour (Figure 3b).

Histopathology
Histopathological assessment revealed a spindle cell neoplasm composed of densely packed fascicles, sometimes wavy in appearance (Figure 2a).  Tumour cells were elongated, with hyperchromatic nuclei, lacking significant mitotic activity.  The fascicles in areas appeared separated by bands of eosinophilic collagen.  On immunohistochemistry, the tumour cells were positive for CD34 (Figure 2b), Bcl-2 (Figure 2c) and focally for CD99 (Figures 2d). Proliferation index with Ki67 was not significantly raised.  Variable reactivity for smooth muscle actin was seen, but the tumour was immunonegative for EMA, S-100 protein, neurofilament and desmin.  Overall, the appearances were typical of a benign SFT (Figure 2).

Discussion
Several techniques now exist to access tumours predominantly within the confines of the sinonasal cavity and the anterior skull base, with broad categorisation into open and endoscopic approaches.  Traditional open techniques, including lateral rhinotomy, craniofacial resection, midface degloving and subfrontal craniotomy, are all associated with significant morbidity to the patient with increased length of hospital stay.  By contrast, the more recent endoscopic trans-nasal approach is associated with reduced surgical trauma, morbidity and a shorter post-operative recovery ,  Benefits for the surgeon include better optics (ie better illumination and wider angle of view) and improved surgical access, although there is an operative learning curve with this newer technique10,.

In coblation, electrolytes in a conductive medium (ie normal saline) are excited by bipolar radiofrequency creating precisely focused plasma. Such plasma-forming settings are used for the ablation of tissues while non-plasma-forming settings can be applied for shrinkage of tissues and haemostasis, thus allowing ablation of tissues at lower temperatures reducing the level of collateral damage to adjacent structures. As a surgical technique coblation enables the operator to cut and coagulate at the same time. Within the field of ENT surgery, it is currently licensed for use in tonsillectomy, adenoidectomy, turbinate resection, partial glossectomy, polypectomy and uvulopharyngopalatoplasty.  Moreover, in a case series of 23 patients, where the use of coblation was compared to traditional operative techniques, it was found that coblation devices were associated with a significant decrease in blood loss (350ml vs. 1000ml). A more recent case series reported on the use of coblation for 15 sinus tumours, including its use on 4 vascular tumours6. It was found to be a very effective tool for such resections and the authors described a low level of blood loss of <400ml on average.

In the present case, the use of the endoscopic approach combined with coblation enabled macroscopic resection of a vascular tumour, with minimal blood loss and an excellent intra-operative field of view. To the best of our knowledge our case is the first reported use of coblation to resect a sinonasal SFT. 

To our knowledge, there have been 35 published cases of sinonasal SFTs, differing in location and size, but with nasal obstruction being the commonest presenting symptom-33. Other presentations included epistaxis, anosmia and headache. The mainstay of management of such cases is operative with radiotherapy reserved for incomplete resection. Operative technique varied and there were 14 endoscopic and, 12 open (majority lateral rhinotomy and /or craniofacial resection) surgical approaches15-33. In 9 cases the mode of resection was not documented19,25,27. Attempt at endoscopic removal led to significant amounts of bleeding in one case leading to a secondary open approach being used24. Length of follow-up varied between 3 months to 12 years and only 2 of the 31 patients that were followed up required additional management after the primary surgery. In one such case the patient was given radiotherapy and the second underwent a completion resection via an open approach, having failed to clear the disease endoscopically. In the present case, despite the reassuring early post-operative MR scan, long-term clinical and radiological follow-up is mandated.

It was once advocated that the gold standard for access to anterior skull base disease is with an open approach. However, with the emerging experience with endoscopic techniques, there may be a shift towards more minimally invasive approaches and as such, discovering new techniques such as coblation to help surgeons is vital.DECLARATION
R Edmiston wrote the paper.  P Pal reviewed the histology.  K Gnanalingham jointly managed the surgical care of the patient and revised the manuscript.  RK Bhalla provided editorial advice and was responsible overall for patient care.  RK Bhalla accepts full responsibility for the integrity of the article.

FINANCIAL INTERESTS:  
Arthrocare provided a financial contribution to the 4th Manchester Sinus, Rhinoplasty and Nasal Plastics Dissection Course in June 2013.

CONFLICTS OF INTEREST:  
None.






SUMMARY: 
Sinonasal solitary fibrous tumours (SFT) are extremely rare tumours of mesenchymal origin 
Their resection can be complicated by difficulty with anatomical access and increased tumour vascularity.
In the present case of recurrent sinonasal SFT involving the anterior skull base, a macroscopic resection was feasible using an endoscopic approach with the use of coblation.
Coblation, which is as yet unreported in the literature for sinonasal SFTs, was found to be an effective means of ensuring haemostasis whilst maintaining an excellent field of view.











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