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Journal of Indian Society of Pedodontics and Preventive Dentistry Official publication of Indian Society of Pedodontics and Preventive Dentistry
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Year : 2007  |  Volume : 25  |  Issue : 1  |  Page : 49-51

Reactivation of trigeminal neuralgia following distraction osteogenesis in an 8-year-old child: Report of a unique case

1 Department of Cleft and Craniofacial Surgery, Meenakshi Ammal Dental College and Hospital, Chennai, India
2 Department of Oral and Maxillofacial Surgery, Meenakshi Ammal Dental College and Hospital, Chennai, India

Correspondence Address:
N Jayakumar
1, 5th Street, Dr. Subbrayan Nagar, Kodambakkam, Chennai - 600 024
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/0970-4388.31992

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Trigeminal neuralgia is extremely rare in children. No concrete treatment protocols seem to be available for management of this condition in the pediatric population. Although trigeminal neuralgia may achieve remission, the possibility of reactivation of a hitherto quiescent condition cannot be ruled out. We present a case of pediatric trigeminal neuralgia following distraction osteogenesis of the mandible.

Keywords: Distraction osteogenesis, hemifacial microsomia, pediatric trigeminal neuralgia, romberg′s disease

How to cite this article:
Ramanathan M, Parameshwaran AA, Jayakumar N, Raghaviah A M. Reactivation of trigeminal neuralgia following distraction osteogenesis in an 8-year-old child: Report of a unique case. J Indian Soc Pedod Prev Dent 2007;25:49-51

How to cite this URL:
Ramanathan M, Parameshwaran AA, Jayakumar N, Raghaviah A M. Reactivation of trigeminal neuralgia following distraction osteogenesis in an 8-year-old child: Report of a unique case. J Indian Soc Pedod Prev Dent [serial online] 2007 [cited 2022 Dec 1];25:49-51. Available from: http://www.jisppd.com/text.asp?2007/25/1/49/31992

   Introduction Top

Manifestation of trigeminal neuralgia in children is almost similar to that in adults but for certain aspects, making it clearly a challenge in pediatric cranio-facial practice. Specific features of pediatric neuralgia include scarcity of pain free episodes and an exaggerated debilitation leading to performance lack in essential activities. [1],[2] With regard to management, non-surgical methods have produced a varying degree of pain relief in children. [3] The inherent side effects of drugs used in trigeminal neuralgia have seldom been tolerated well by children. Nevertheless surgical options have their own disadvantages too. Microvascular decompression is a more complex surgery to be performed in children than in adults because of the involvement of varying number of vessels. [4],[5],[6] Children also seem to take a longer time to recover following the procedure. [6],[7] The most favored gamma knife surgery needs well-defined indications to perform in children considering the amount of radiation. [8],[9] Also the inherent complications following glycerol rhizotomies like facial and corneal sensory loss, keratitis, trigeminal motor weakness, reactive meningitis, narrowing of the cerebrospinal cistern should also be considered before opting for the procedure. Some authors advocate use of peripheral glycerol injections as a safe procedure with minimal side effects in children. [10],[11],[12]

   Case Report Top

An 8-year-old female reported to the Department of Cleft and Cranio-facial surgery, Meenakshi Ammal Dental College and Hospital, Chennai, India with a complaint of facial disfigurement. Patient presented with a gross asymmetry in the form of a unilateral maxillo-mandibular hypoplasia on the right side. History revealed an episode of vesicular lesions on the affected side with associated fever at her 5 th year of life. These episodes were apparently self-limiting. The parents noticed significant growth retardation following this event.

Clinically, the child exhibited classical signs of Parry Romberg syndrome including hypoplasia of mandible, infra-orbital rims, and malar prominence along with marked soft tissue deficit and muscular atrophy [Figure - 1].

Routine radiographic and computerized tomography evaluation were suggestive of a Type I Pruzansky's Mandibular deformity.

An extra-oral monofocal distractor device was fixed under general anesthesia [Figure - 2] as a part of the treatment plan to correct the mandibular hypoplasia. The surgery was uneventful.

The post-operative period was not significant for the first 8 hours following which patient complained of severe radiating unilateral hemifacial pain, which was throbbing in nature. A diagnosis of post-operative pain was made and the patient was given NSAIDS. Since the pain persisted with the same intensity and did not respond to treatment, a more detailed history of the nature of pain on the second post-operative day was done revealing paroxysmal lancinating pain lasting 1-2 minutes with pain free intervals of 20 minutes.

At this juncture the patient's mother revealed a history of similar episodes 2 years back. This information was concealed pre-operatively and on further probing revealed that the patient was diagnosed earlier with Trigeminal Neuralgia at a specialty neurology center and was on a prescription of carbamazepine for a period of 3 months. Upon remission of symptoms the drug regimen has been discontinued against medical advice.

A fresh neurological evaluation was obtained and a diagnosis of Atypical Trigeminal Neuralgia was arrived. The patient was put on a prescription of Clonazepam 5 mg by oral route 12 hourly and Carbamazepine 200 mg 12 hourly. This regimen was to be continued for a period of 2 weeks before further review. Three days after the drug therapy was started the patient experienced complete remission of all symptoms and was totally pain free even during the activation phase for the distraction device. The patient was reviewed at intervals of 2 weeks for a period of 3 months and is completely symptom free. The distraction device removal was successfully done under local anesthesia without exacerbation of any symptoms.

   Discussion Top

Trigeminal neuralgia is a complex neurological entity predominantly seen in the 4 th and 5 th decades of life. [13] The very manifestation of this disorder in the pediatric population is a rarity. The true cause of this condition is still debatable but a widely accepted mechanism proposed initially by Dandy followed by Gardener and redefined by Janetta as de-myelination caused by vascular compression of the trigeminal root adjacent to the pons- dorsal root entry zone. [14],[15] In general, the diagnosis of TN is based on a history of characteristic pain attacks that are consistent with specific widely accepted research and clinical criteria for diagnosis. [16],[17] More detailed investigations and adoption of Sweet's criteria may help in confirmation. [16]

In children, facial asymmetry caused by unilateral hypoplasia of mandible is either congenital (hemifacial microsomia) or acquired. [18] Hemifacial microsomia is a relatively common cranio-facial anomaly with an incidence of 1 per 3500 live births (Poswillo). [19] It is characterized by unilateral under development of mandible, maxilla, zygoma, external and middle ear, parotid gland, fifth, and seventh cranial nerves and associated musculature and soft tissues. [19] Extra cranio-facial anomalies that can be found in conjunction with hemifacial microsomia include central nervous system, skeletal, and cardiac malformations. [20] Correlation between trigeminal neuralgia and congenital facial anomalies (in this case Romberg's disease) carries very less attention in literature. Though reactivation of latent TN is less understood, a possible mechanism has been proposed by Ecker and Smith. [21] They suggest that the primary cause may be a single, active DNA sequence in the persistent but non-integrated genome of latent herpes simplex virus type 1 commonly observed in a few infected A-delta nerve fibers of the cheek. Hence facial pain may occur following reactivation of this virus. We believe that the surgical procedure of distraction osteogenesis in the affected side may be an important predisposing factor for the recurrence as observed. The surgical insult due to osteotomy and a possible nerve injury may be contributory factors. Intra-operative activation of the device to confirm distraction may have produced undue nerve traction thereby leading to retriggering of the pathology.

   Conclusion Top

It is the inference of the authors that any form of interventional procedures in a patient with pre-existing TN may precipitate or aggravate a latent or quiescent lesion. Planning surgical procedures in such patients needs to be done with caution.

   References Top

1.Lopes PG, Castro ES Jr, Lopes LH. Trigeminal neuralgia in children: Two case reports. Pediatr Neurol 2002;26:309-10.   Back to cited text no. 1  [PUBMED]  [FULLTEXT]
2.Dangel T. Chronic pain management in children. Part I: Cancer and phantom pain. Paediatr Anaesth 1998;8:5-10.  Back to cited text no. 2    
3.Yue WL. Peripheral glycerol injection for the relief of facial pain in children. Int J Pediatr Otorhinolaryngol 2004;68:37-41.   Back to cited text no. 3  [PUBMED]  [FULLTEXT]
4.Li ST, Pan Q, Liu N, Shen F, Liu Z, Guan Y. Trigeminal neuralgia: What are the important factors for good operative outcomes with Microvascular decompression. Surg Neurol 2004;62:400-5.  Back to cited text no. 4  [PUBMED]  [FULLTEXT]
5.Hatayama T. Intraoperative mapping of the trigeminal nerve root during MVD. Int Congr Ser 2004;1282:347-52.  Back to cited text no. 5    
6.Kondo A, Tanabe H. What should be done, what should not be done in microvascular decompression surgery. Int Congr Ser 2004;1282:353-6.  Back to cited text no. 6    
7.Resnick DK, Levy EI, Jannetta PJ. Microvascular decompression for pediatric onset trigeminal neuralgia. Neurosurgery 1998;43:804-8.  Back to cited text no. 7  [PUBMED]  [FULLTEXT]
8.Shaya M, Jawahar A, Caldito G, Sin A, Willis BK, Nanda A. Gamma Knife Radiosurgery for trigeminal neuralgia: A study of predictors of success, efficacy, safety and outcome at LSUHSC. Surg Neurol 2004;61:529-35.   Back to cited text no. 8  [PUBMED]  [FULLTEXT]
9.Lindquist C. Gamma Knife Radiosurgery. Semi Radiat Oncol 1995;5:197-202.   Back to cited text no. 9  [PUBMED]  
10.Stajcic SS. Peripheral glycerol injection for the relief of trigeminal neuralgia. Int J Oral Maxillofac Surg 1989;18:255-7.   Back to cited text no. 10    
11.Yue WL, Li Y, Zhang JC. Peripheral glycerol injection for relief of tic douloureux. J Otolaryngol (Chin) 1989;18:23-5.   Back to cited text no. 11    
12.Zakrzewska JM. Trigeminal neuralgia and facial pain. Semi Pain Med 2004;2:204-14.   Back to cited text no. 12    
13.Lunsford LD, Bennett MH. Percutaneous retrogasserian glycerol rhizotomy for tic douloureux. Part I. Neurosurgery 1984;14:424-9.   Back to cited text no. 13    
14.Apfelbaum RI. Comparison of the long-term results of microvascular decompression and percutaneous trigeminal neurolysis for the treatment of trigeminal neuralgia. International Congress Series 2002;1247:629-43.   Back to cited text no. 14    
15.Zakrewska JM. Trigeminal neuralgia and facial pain. Semi Pain Med 2004;2:76-84.   Back to cited text no. 15    
16.Scrivani SJ, Mathews ES, Maciewicz RJ. Trigeminal neuralgia. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2005;100:527-38.   Back to cited text no. 16  [PUBMED]  [FULLTEXT]
17.Zakrzewska JM. Diagnosis and differential diagnosis of trigeminal neuralgia. Clin J Pain 2002;18:14-21.  Back to cited text no. 17  [PUBMED]  [FULLTEXT]
18.Jansma J, Bierman MW, Becking AG. Intraoral distraction osteogenesis to lengthen the ascending ramus. Experience with seven patients. Br J Oral Maxillofac Surg 2004;42:526-31.   Back to cited text no. 18    
19.Meazzini MC, Mazzoleni F, Gabriele C, Bozzetti A. Mandibular distraction osteogenesis in hemifacial microsomia: Long-term follow-up. J Craniomaxillofac Surg 2005;33:370-6.   Back to cited text no. 19  [PUBMED]  [FULLTEXT]
20.Trahar M, Sheffield R, Kawamoto H, Lee HF, Ting K. Cephalometric evaluation of the craniofacial complex in patients with an intraoral distraction osteogenesis device: A preliminary report. Am J Orthodont Dentofac Orthop 2003;124:639-49.   Back to cited text no. 20  [PUBMED]  [FULLTEXT]
21.Ecker AD, Smith JE. Are latent, immediate-early genes of herpes simplex virus-1 essential in causing trigeminal neuralgia? Med Hypotheses 2002;59:603-6.  Back to cited text no. 21  [PUBMED]  [FULLTEXT]


  [Figure - 1], [Figure - 2]

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