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 Table of Contents  
CASE REPORT
Year : 2016  |  Volume : 30  |  Issue : 1  |  Page : 67-69

An unusual presentation of trigeminal neuralgia caused by fibrous dysplasia (managed with radiofrequency lesioning)


Department of Anaesthesiology, Pain Management Center, Kamineni Hospitals, King Koti, Hyderabad, Telangana, India

Date of Web Publication7-Jan-2016

Correspondence Address:
Dr. Neha Kanojia
Department of Anaesthesiology, Pain Management Center, Kamineni Hospitals, King Koti, Hyderabad, Telangana
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0970-5333.173483

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  Abstract 

Fibrous dysplasia (FD) is a skeletal developmental disorder of the unknown etiology, uncertain pathogenesis, and diverse histopathology with one-fourth involving head and neck. The aim of this article is to report a rare case of craniofacial FD of the maxillary sinus as an etiology of trigeminal neuralgia treated with radiofrequency lesioning.

Keywords: Fibrous dysplasia, radiofrequency lesioning, trigeminal neuralgia


How to cite this article:
Joshi M, Kanojia N, Rao UW, Basavaraju L. An unusual presentation of trigeminal neuralgia caused by fibrous dysplasia (managed with radiofrequency lesioning). Indian J Pain 2016;30:67-9

How to cite this URL:
Joshi M, Kanojia N, Rao UW, Basavaraju L. An unusual presentation of trigeminal neuralgia caused by fibrous dysplasia (managed with radiofrequency lesioning). Indian J Pain [serial online] 2016 [cited 2020 May 31];30:67-9. Available from: http://www.indianjpain.org/text.asp?2016/30/1/67/173483


  Introduction Top


Fibrous dysplasia (FD) is a skeletal developmental disorder of the bone-forming mesenchyme that manifests as a defect in osteoblastic differentiation and maturation. It is a benign bone disorder of uncertain pathogenesis, and diverse histopathology. Cranial or facial bones are affected approximately in 30% of the patients. [1],[2] Although the maxilla and mandible are most commonly involved, maxillary sinus involvement is rare and furthermore, rarely involves foramen ovale (FO) consisting trigeminal nerve. [2] Because of involvement of trigeminal nerve it can mimic trigeminal neuralgia (TN).

TN or tic douloureux is defined as paroxysmal and/or recurrent attack of pain lasting from a fraction of second to few minutes, involving one or more divisions of fifth cranial nerve. [3] Onset is usually after 40 years of age with a peak occurrence between 50 and 80 with a female preponderance (female/male ratio- 1.74:1). TN is twice as common on the right side than left side. [3],[4]


  Case Report Top


A 35-year-old female with no significant medical history was referred to the Department of Anaesthesia and Pain Medicine with a history of lancinating pain along left maxillary region since 5 years. The patient was a known case of FD of left maxilla and was managed surgically 2 years back and was confirmed on histopathology to be osteofibrous hyperplasia.

The patient was prescribed antineuropathic drugs such as carbamazepine, pregabalin, and others, but she did not have adequate pain relief. The patient became pregnant during this time, and the drugs were discontinued. In the third trimester of pregnancy, the patient had severe pain and was managed with infraorbital nerve block with local anesthesia. The patient received infraorbital block 3 times during pregnancy and lactation period and had good pain relief for few months and was planned for radiofrequency (RF) lesioning of V2 division at a later date.

Pre-anesthetic workup for RF lesioning of V2 division of TN was done. Routine investigations along with special investigations such as X-ray PNS, X-ray skull, MRI Brain, PET CT Scan and 3D CT Scan were done to know the extent of FD [Figure 1] and displacement of FO [Figure 2]). The reports are in [Table 1].
Figure 1: Three-dimensional computed tomography scan showing fibrous dysplasia involving maxillary sinus


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Figure 2: Lytic expansile lesion at greater wing of sphenoid on the left side medial and lateral pterygoid plates on left side and skull base (arrow showing the displacement of foramen ovale from normal)


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Table 1: Special investigations


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Procedure

The patient was positioned in the supine position with neck slightly extended. After prophylactic antibiotic, standard hemodynamic parameters were monitored every 5 min until the end of the procedure. Fluoroscopic guidance was used to confirm the position of FO and to determine the direction of the RF needle. The fluoroscope was positioned to get an occipito-submental view (water's view). A 15-20° tilt to the ipsilateral affected side was given to improving visualization of the FO. The FO could not be visualized clearly in view of overlapping maxillary FD. We used a 10 cm 20G RF needle with a 5 mm straight uninsulated tip. The needle entry point was midpoint of FO, which turned out to be 4 cm from the corner of the mouth. Under all aseptic precautions, the skin over the needle entry point was anesthetized with 1% lidocaine. Once we aligned needle to FO and achieved desired depth, sensory stimulation was carried out at 50 Hz and motor stimulation done at 2 Hz. The definitive position of the electrode was verified by inducing paresthesia as expressed by the patient with sensory stimulation between 0.1 and 0.3 V in the affected painful area. The patient was given intravenous sedation with injection propofol 1 mg/kg and injection fentany l 2 mcg/kg during RF lesioning. The RF lesion was done for 3 times at the level of clivus at 70° for 90 s each. On awakening from sedation, the patient had complete pain relief and numbness over V2 division of trigeminal nerve.


  Discussion Top


This is a rare case of maxillary sinus FD-causing narrowing of the FO leading to TN. As we know by now that FD is a benign bone disorder of an unknown etiology, uncertain pathogenesis and diverse histopathology, [1],[2] TN is a severe, almost exclusively unilateral, neuropathic pain located within the distribution of the fifth nerve manifesting as paroxysmal high-intensity jabs or stabs lasting seconds. [3],[5]

FD rarely affects the head and neck region. Moreover, the involvement of the paranasal sinuses is rare. Although the maxilla and mandible are most commonly involved, maxillary sinus involvement is rare. [6],[7]

Classically TN is managed with drug therapy. Other treatment modalities such as peripheral nerve blocks and surgical options - microvascular decompression, stereotactic radiosurgery, percutaneous microcompression rhizolysis are reserved for cases refractory to medical management. [8],[9] RF lesioning has become as an alternative therapy to avoid the adverse effects associated with medications and invasive surgical procedures. [10],[11] It is safe, efficacious minimally invasive and target selective nature. In this case, performing radio frequency lesioning was difficult as the localization of the FO was difficult due to the anatomical distortion by the FD, which could be managed with good imaging technique. However, the patient had complete pain relief following the RF lesioning and discharged with a prescription.


  Conclusion Top


FD is a nonneoplastic lesion of unknown origin involving head and neck. This is a rare case of FD of the maxillary sinus as an etiology of trigeminal neuralgic pain successfully treated with RF lesioning.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
  References Top

1.
Cholakova R, Kanasirska P, Kanasirski N, Chenchev Iv, Dinkova A. Fibrous dysplasia in the maxillomandibular region - Case report. J IMAB - Annu Proc (Scientific Papers) 2010;16(book 4).  Back to cited text no. 1
    
2.
Subramaniam V, Herle AT. Fibrous dysplasia of the maxillary sinus: Case report. South Brazilian Dentistry Journal 2010;7:366-8.  Back to cited text no. 2
    
3.
Joffroy A, Levivier M, Massager N. Trigeminal neuralgia. Pathophysiology and treatment. Acta Neurol Belg 2001;101:20-5.  Back to cited text no. 3
    
4.
Joshi M. Headache. In: Textbook of Pain Management. 3 rd ed. Hyderabad: Paras Medical Publisher; 2014. p. 180-92.  Back to cited text no. 4
    
5.
Grasso G, Passalacqua M, Giambartino F, Cacciola F, Caruso G, Tomasello F. Typical trigeminal neuralgia by an atypical compression: Case report and review of the literature. Turk Neurosurg 2014;24:82-5.  Back to cited text no. 5
    
6.
Ben hadj Hamida F, Jlaiel R, Ben Rayana N, Mahjoub H, Mellouli T, Ghorbel M, et al. Craniofacial fibrous dysplasia: A case report. J Fr Ophtalmol 2005;28:e6.  Back to cited text no. 6
    
7.
Ozek C, Gundogan H, Bilkay U, Tokat C, Gurler T, Songur E. Craniomaxillofacial fibrous dysplasia. J Craniofac Surg 2002;13:382-9.  Back to cited text no. 7
    
8.
Zakrzewska JM, Akram H. Neurosurgical interventions for the treatment of classical trigeminal neuralgia. Cochrane Database Syst Rev 2011;7:CD007312.  Back to cited text no. 8
    
9.
Nurmikko TJ, Eldridge PR. Trigeminal neuralgia - pathophysiology, diagnosis and current treatment. Br J Anaesth 2001;87:117-32.  Back to cited text no. 9
    
10.
Waldmann SD. Trigeminal nerve block. In: Weiner RS, editor. Innovations in Pain Management. Vol. 1. Orlando, FL: PMD Press; 1990. p. 10-5.  Back to cited text no. 10
    
11.
Katz J, editor. Gasserian ganglion. In: Atlas of Regional Anesthesia. Norwalk, CT: Appleton and Lange; 1994. p. 4-5.  Back to cited text no. 11
    


    Figures

  [Figure 1], [Figure 2]
 
 
    Tables

  [Table 1]



 

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