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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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CASE REPORT
Year : 2017  |  Volume : 35  |  Issue : 1  |  Page : 90-93
 

Management of tongue and lip laceration due to dystonia in a 1-year-old infant


Department of Pedodontics and Preventive Dentistry, Cloudnine Kids Children's Hospital, AECS Maaruti College of Dental Sciences and Research Centre, Bengaluru, Karnataka, India

Date of Web Publication31-Jan-2017

Correspondence Address:
J P Beena
47, 9th Cross, 29th Main, Ist Phase, J. P. Nagar, Bengaluru - 560 078, Karnataka
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0970-4388.199223

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   Abstract 

This case report describes the management of tongue and lip lacerations due to dystonia in a 1-year-old infant. A splint was given to raise the bite and prevent repeated trauma and aid in healing of the oral tissue. This paper highlights the importance of pediatric dentist's role in improving quality of patient care in an intensive care unit.


Keywords: Dystonia, infant, tongue laceration


How to cite this article:
Beena J P. Management of tongue and lip laceration due to dystonia in a 1-year-old infant. J Indian Soc Pedod Prev Dent 2017;35:90-3

How to cite this URL:
Beena J P. Management of tongue and lip laceration due to dystonia in a 1-year-old infant. J Indian Soc Pedod Prev Dent [serial online] 2017 [cited 2017 Apr 26];35:90-3. Available from: http://www.jisppd.com/text.asp?2017/35/1/90/199223



   Introduction Top


Dystonia (from Greek, meaning altered muscle tone) describes a syndrome of involuntary sustained or spasmodic muscle contractions involving co-contraction of the agonist and the antagonist. The earliest account of dystonia dates back to around 1911. These movements are generally slow, sustained, and are often in a repetitive and patterned manner. They can be unpredictable and fluctuate in nature as well.[1]

Dystonias are classified according to the following characteristics:[1]

  1. Age of onset
  2. Etiology
  3. Anatomic distribution.



   Classification by Age of Onset Top


With regard to patient age, dystonias can be classified as follows:

  • Infantile dystonia - Begins before age 2 years
  • Childhood dystonia - Begins at age 2–12 years
  • Juvenile dystonia - Begins at age 13–20 years
  • Adult dystonia - Begins after age 20 years.



   Etiologic Classification Top


Primary (idiopathic) dystonia

Primary or idiopathic, dystonias may be present in a sporadic, autosomal dominant, autosomal recessive, or X-linked recessive manner. Heritable childhood-onset dystonia is particularly common among Ashkenazi Jews.

Secondary dystonia

This may result from a wide variety of neurologic diseases or inherited metabolic defects, including the following: Huntington disease,  Hallervorden-Spatz disease More Details, Wilson disease (hepatolenticular degeneration), Leigh disease, Lipid storage disease,  Parkinsonism More Details, Central nervous system infections, Cerebral or cerebellar tumors. Other causes include drug intoxication from Dopamine antagonists, neuroleptics, metoclopramide, and haloperidol. Structural or hypoxic injury to the basal ganglia brainstem structures could also lead to dystonia.


   Anatomic Classification Top


On the basis of its clinical distribution, dystonia is classified as follows:

  • Focal dystonia-Involving a single body part
  • Segmental dystonia - Affects two or more contiguous regions of the body; example: of segmental dystonias of the head and neck include craniocervical dystonia, blepharospasm, oromandibular dystonia, and laryngeal dystonia.
  • Multifocal dystonia - Consists of abnormalities in noncontiguous body parts
  • Generalized dystonia - Involves segmental crural dystonia and at least one other body part; e.g.: dystonia musculorum deformans (or torsion dystonia), a generalized form of the disease, which involves the trunk and limbs.[2]
  • Hemidystonia - Also called unilateral dystonia; usually associated with abnormalities in the contralateral basal ganglia.[3]


Case report

Medical history

An 1 year old male child was admitted with a complaint of Dystonic posturing since 10 days and fever since 1 week which was high grade and intermittent.

Past medical history

Child was apparently normal till 7 months age when he started having regression of milestones in the form of loss of neck control, difficulty to sit/or hold objects, difficulty in swallowing food and reduced activity which were present before this age. For above complaints, child was currently being evaluated at Neurological Institute for Mitichondrail Encephalopathy/? leigh disease/? organic aciduria with acute dystonia/? sepsis? and genetic studies were sent (reports waited).


   Course in the Hospital Top


Child was brought to Emergency Room in Dystonic state, when IV Loraz (Lorazepam) was given stat with which the child settled. Child was managed with IV fluids and antibiotics. Child had high C-reactive protein levels with high counts, was started on IV antibiotics (piptaz-piperacillin/tazobactam). Blood culture was sent. Fever spikes came down with the above treatment. For dystonia Paciten 2mg twice daily(Trihexyphenidyl) was administered. On the 3rd day of admission child again had severe dystonia which settled with IV Loraz stat. Child was started on Clonazepam 5mg once daily. Meanwhile the NGT (Naso gastric tube) feeds were started and the mother was taught the same due to intermittent episodes of dystonia. Child improved and was not having any fever spikes, blood culture showed no growth. Child tolerated the feeds well and was planned to shift to Neurological Institute for further follow up.


   Dental History Top


Chief complaint

Profuse intraoral bleeding was noticed repeatedly by the PICU (Pediatric intensive care unit) staff in the patient and the Pediatric Dentist was notified to examine the patient.

The patient was referred to the pediatric dentist on the last day before discharge.

Extra oral examination

Oval shaped ulcerated area in the lower lip was noted which was yellowish to pale white in colour measuring approximately 4cm x 4cm. The borders were well defined, margins of the ulcer at places were fluctuant and appeared to be filled with clear fluid. The other margins were irregular. Submandibular and submental lymph nodes were not palpable.

Intraoral examination

Teeth present:

52 51 61 62

82 81 7172

Deep ulceration was seen on the ventral surface of the tongue and floor of the mouth with irregular margins, yellowish to pale white in color measuring approximately 6 cm × 5 cm.

Bilateral ulcerations were also seen on the alveolar ridges in posterior molar region of the mandible [Figure 1].
Figure 1: Traumatic ulcerations of the tongue and lower lip due dystonia

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Provisional diagnosis

Traumatic ulcerations of the tongue and lower lip due dystonia.

Treatment plan

Topical anesthetics were prescribed to alleviate the pain.

Because of the age of the patient, decision was made to give a splint to raise the bite Splint would reduce the chances of patient occluding the teeth and hence give the ulceration time to heal. The patient was getting discharged the next day.


   Procedure Top


Upper and lower jaw impressions were recorded, and casts poured. Custom made splint was fabricated for the upper jaw with slightly raised bite in the molar region [Figure 2]. The splint was stabilized with the dental floss to assist the parents in removing and cleaning the appliance [Figure 3].
Figure 2: Custom made splint was fabricated for the upper jaw with slightly raised bite in the molar region

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Figure 3: The splint was stabilized with the dental floss to assist the parents in removing and cleaning the appliance

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Patient was scheduled for a follow up check up in a week's time to check for healing and any readjustment of the splint if needed.


   Discussion Top


Physical restraint aids in a individualized manner to prevent self injury e.g in the case of lesions owing to biting.[4] Intraoral devices work through two mechanisms: they hinder the patient from indulging in self-injurious habits, thus limiting this behavior. Second, they act as a direct barrier, thus preventing injuries to oral tissues. It has been suggested that in order for the intraoral devices to be successful in preventing self-injury, they should accomplish the following, i.e., they should maintain the injured tissues at a distance from the dental arches, so that they do not experience further injury; they must not aggravate newer lesions; they should permit full movement of the mandible; they should not interfere with routine oral hygiene; they should be easy to fabricate and insert into the mouth, without causing further discomfort to the patient; they should aid in healing of the oral tissues and also withstand the forces exerted by the oral structures without being displaced or breaking.[5],[6],[7]

No intraoral devices are free of adverse effects; they hamper oral hygiene, could favor fungal infections and can incite new lesions. Regular readjustments may be required, and the fabrication may require time to produce the devices thus limiting their application in urgent situations.[8]


   Conclusion Top


We managed an infant with lip and tongue lacerations the following dystonia in an intensive care unit using a splint. There are very limited data regarding dental management of dystonia, especially in infants. The limitation of this case report includes the lack of follow-up as the patient was being shifted to another hospital and they were from neighboring country and had to return the following week. Through this paper, we would like to contribute toward awareness among various medical professionals and general dentists, for early recognition of oral injuries and prompt referral to a pediatric dentist to limit the severity of damages to the oral and dental tissues. Interprofessional collaborations between pediatricians and pediatric dentist can lead to better patient care and recovery.

Acknowledgment

The author would like to thank the staff of Cloudnine Kids Children's Hospital, Bengaluru, Karnataka, India.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
   References Top

1.
Fahn S, Bressman SB, Marsden CD. Classification of dystonia. Adv Neurol 1998;78:1-10.  Back to cited text no. 1
    
2.
Hornykiewicz O, Kish SJ, Becker LE, Farley I, Shannak K. Biochemical evidence for brain neurotransmitter changes in idiopathic torsion dystonia (dystonia musculorum deformans). Adv Neurol 1988;50:157-65.  Back to cited text no. 2
    
3.
Alkhani A, Bohlega S. Unilateral pallidotomy for hemidystonia. Mov Disord 2006;21:852-5.  Back to cited text no. 3
    
4.
Saemundsson SR, Roberts MW. Oral self-injurious behavior in the developmentally disabled: Review and a case. ASDC J Dent Child 1997;64:205-9, 228.  Back to cited text no. 4
    
5.
Yasui EM, Kimura RK, Kawamura A, Akiyama S, Morisaki I. A modified oral screen appliance to prevent self-inflicted oral trauma in an infant with cerebral palsy: A case report. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2004;97:471-5.  Back to cited text no. 5
    
6.
Kiat-Amnuay S, Koh SH, Powner DJ. An occlusal guard for preventing and treating self-inflicted tongue trauma in a comatose patient: A clinical report. J Prosthet Dent 2008;99:421-4.  Back to cited text no. 6
    
7.
Hanson GE, Ogle RG, Giron L. A tongue stent for prevention of oral trauma in the comatose patient. Crit Care Med 1975;3:200-3.  Back to cited text no. 7
    
8.
Cusumano FJ, Penna KJ, Panossian G. Prevention of self-mutilation in patients with Lesch-Nyhan syndrome: Review of literature. ASDC J Dent Child 2001;68:175-8.  Back to cited text no. 8
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]



 

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    Abstract
   Introduction
    Classification b...
    Etiologic Classi...
    Anatomic Classif...
    Course in the Ho...
   Dental History
   Procedure
   Discussion
   Conclusion
    References
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