Drug Therapies of Bruxism in Down Children: Preliminary Report

Achille Lamma, MD & Renato Cocchi M.D., Ph.D. (Sociology)
Italian Journal of Intellective Impairment 1 (1): 19-24 (1988)

Reprinted with the permission of Renato Cocchi
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Summary

134 home reared Down's syndrome children (95 M + 49 F; age range at 1st consultation: 5/12 - 15 + 8/12 years; average = 5 + 11/12; 133 normally distributed chromosomal cytodiagnoses) presented bruxism on 1st consultation or on one of the subsequent checkups, when they had had the 1st visit before they were one year old. The administered drug therapies, chiefly aimed at reducing stress reactions, and summarized into 9 therapeutic frameworks, did eliminate this symptom in 62.68% Ss and mitigate it in a further 29.85%, after 8-22 months treatment.
The possible specific role of some drugs used as well as their synergisms are discussed in connection with the results.
Key words: Bruxism; Down's syndrome; drug therapy.

While continuing to concern ourselves with a subject which we have already researched into (Cocchi and Lamma, 1987; Lamma, 1987), it was decided to evaluate, in a rather more systematic manner, the effect of drug therapies on bruxism. By drug therapies we do not mean just any kind of therapy, but only the type which is applied in Pesaro and which is chiefly aimed at reducing reactions to stress (Cocchi, 1987).

This is based on the premise that the Down individual, from his day of conception, is subjected to a metabolic endogenous stress as the result of the acceleration of all the metabolisms, the enzymes of which depend for their controlling genes no more on two but on three chromosome 21 (Cocchi, 1987).

The fact that the therapy is not standard but as far as possible tailored to the presumed needs of each child, and that the space available is limited, do not allow for more than an overall presentation requiring further deeper statistical refinements, already in progress.

Materials and methods.

From a non-selected, consecutive series of medical records referring to 408 home reared Down subjects representing all areas of Italy, and all examined by one of us between 1979 and April 1988, 134 were eligible for this investigation.

The selection criteria were as follows:

the identification of bruxism on the first examination or on one of the successive checkups if the child was less than one year old at the first visit;
annotation of the intensity of the symptom on both first and last examination, after a period of undertaking the prescribed drug therapy.

From the 134 records selected on the above criteria, the following data were taken:

sex.
age at first consultation;
chromosomal diagnosis;
initial (scale: ½; ¾; 1; 2) and final intensity (scale: 0; ¼; ½; ¾; 1; 2) of bruxism;
- length of time, in months, between the first and last observation of the symptom, or recording of its disappearance;
- type of drug therapy administered, which fits into one of nine simplified frameworks and so therefore definable:
  1. Basic antistress therapy (BAT) ( l-glutamine or I-glutamine+ pemoline; pyridoxine or pyridoxine+ thiamine or pyridoxine+ thiamine+ cyanocobalamine; sometimes addition of alphatocopherol; a benzodiazepine).
  2. BAT & 5-hydroxytryptophan (5-HTP).
  3. BAT & carbamazepine (CBZ).
  4. BAT & 5-HTP & CBZ.
  5. BAT with successive addition of 5-HTP.
  6. BAT with successive addition of CBZ.
  7. BAT with successive addition of 5-HTP and CBZ
  8. BAT & CBZ with successive addition of 5-HTP.
  9. BAT & 5-HTP with successive addition of CBZ.

The daily dosages, minimum and maximum, have been reported elsewhere (Cocchi, 1997).

Any other type of drug prescribed, for a particular symptom or condition of the child (e.g. autism) have not been taken into consideration in this research.

The results have been interpreted in terms of means and SDs.

The application of Student's t Test for two paired samples on the total results obtained is based on the assumption that the therapies, different in quality, quantity and length of administration, had in some way balanced out any initial inequalities in the subjects reactions to stress.

As this is a debatable assumption, the result of the test can only provide a rough indication.

Results

Sex: 85 M + 49 F; sex ratio = 173.47.
Age at 1^st consultation: range: 5/12 - 15+ 8/12 years; average: 5+ 11/12, with SD = 8 + 3/12 years.
Distribution of chromosomal anomalies:: pure trisomy 21: 121 = 90.29%; translocations: 6 = 4.48%; mosaicisms: 6 = 4.48%; unknown (only clinical diagnosis) 1 = 0.75%

Table 1 shows a summary of the results obtained on bruxism.

Table 1: initial and final scores on bruxism
Intensity grade	Initial scores		Final scores		Difference		Follow-up (months)
Intensity grade	Ss. no.	%	Ss. no.	%	Ss. no.	%	Average ±SD
(0)	0	0.00	84	62.68	f84	+62.68	10.23 ± 7.59
(¼)	0	0.00	10	7.46	f10	+7.46	22.70 ± 14.23
(½)	18	13.43	29	21.64	f11	+8.21	14.17 ± 10.18
(¾)	4	2.99	1	0.75	-3	-2.24	7.92 ± 4.17
(1)	111	82.84	9	6.72	-102	-76.12	19.22 ± 10.51
(2)	1	0.75	1	0.75	0	0.00	25
Totals	134	100.00	134	100.00

t = 26.34 with 133 df; p < .0005 (debatable for possible sample bias)

We felt that the 84 cases in which bruxism disappeared completely were worthy of a more detailed study.
These are represented in Table 2, in progressive order of the average time (in months) in which the symptom disappeared.

Table 2: distribution of the 84 cases in which bruxism completely disappeared, according to therapies and their average time length.
Therapeutic regimen	Ss. no.	%	Score of bruxism		Time in months
Therapeutic regimen	Ss. no.	%	initial	final	Average ± SD
BAT	20	23.81	16	0	6.90 ± 5.91
BAT & 5-HTP & CBZ	3	3.57	3	0	8 ± 4.58
BAT & CBZ	23	27.30	21	0	8.65 ± 7.23
TAP & 5-HTP	13	15.48	11.5	0	8.8 ± 5.88
BAT & 5-HTP+ CBZ	4	4.76	4	0	13 ±4.24
BAT & CBZ+ 5-HTP	1	1.19	1	0	13
BAT+ CBZ	9	10.71	8.75	0	13.11 ± 4.68
TAB+ 5-HTP	7	8.33	6.25	0	18 ± 10.41
BAT+ 5-HTP+ CBZ	4	4.76	4	0	22 ± 9.90
Totals	84	100.00	65.5	0	12.38

Discussion

The number of subjects studied, the distribution of chromosomal cytodiagnoses and the nationwide origin of the probands lead us to believe that this sample must had been representative of the population of Italian Down children affected by bruxism.

A male prevalence had previously been noted (Cocchi and Lamma, 1987) and was indeed born out in these findings.

What also must be pointed out immediately is the large variety of drug combinations: These are dependent on the presence and intensity of the other symtoms observed on the first and successive visits as well as the necessity for caution in cases where the child was very young.

It must also be added that these variations could have been the result of prudence on the part of the therapist especially in the first five years in which these therapies were being tried out and refined.

On the other hand it must be born in mind that while the personalisation and dosages of the drugs prescribed is advantageous to the cure, it does however make any scientific study of the same rather difficult.

Given this, in the current absence of a more refined statistical analysis, the results shown are of a prevalently descriptive value.

In fact we can take it for almost certain that bruxism being a symptom present in many Down subjects and yielding in consequence to therapies based on antistress action, this supports the interpretation, - which is not only ours (Morse, 1982) - that we are dealing with a stress signal in action.

It is also a fact that pharmacological therapy, as carried out in Pesaro, can eliminate this symptom in 62.68 % of cases both by different combinations of a certain number of drugs, as well as administering these at different times.

Until we are able to carry out an analysis of variance (some combinations of drugs have, at the moment, an insufficient number of subjects to do so), we cannot know the reason of different elimination times for bruxism, (from around 7 to 22 months on average). Perhaps these times depend on the symptom itself, within the personal reaction to stress in that particular child and/or on the fact of not having used, from the beginning, all the drugs which could have been useful, or again on still further factors.

As regards drugs, it can be seen that already a basic antistress therapy accounts for almost a quarter of the cured cases.

The other two drugs which, in our opinion, work well for this symptom are CBZ and 5-HPT.

While the choice of CBZ had been made on the basis of its antidystonic properties (Geller, Kaplan and Christoff, 1976; Isgreen et al., 1976), not something widely known by the way, the antibruxism action of 5-HTP is something which originates from this report.

The following can be stated about this action:

—The choice of 5-HTP was not because of bruxism but due to the presence of symptoms of impaired serotonin function:

Bruxism was found in a subject who on the first examination had already been receiving 50 mg per day of 5-HTP for the previous year;
It is possible that the antibruxist action of 5-HTP, if it exists, comes about through an increase in the endogenous synthesis of nicotinamide, the antistress properties of which were reported (Voronina, 1981).

Any speculation as to the most effective drug combinations in dealing with this symptom, on the basis of the results presented here would seem rather hazardous at the moment, even if we are convinced that a better solution could lie in BAT together with CBZ.

Two last collateral observations regarding our research:

As one of us has already noted elsewhere (Lamma, 1987), bruxism, though temporarily eradicated, can return to Down subjects in future times of stress: That is to say during the summer season, in periods of intense sultry heat; when the respiratory tracts are infected, or in the case of exanthematic illnesses, when prolonged antibiotic therapies are needed. This fact has been observed and recorded in several subjects.
We have no experience of the CBZ or 5-HTP as drugs against the bruxism.

Since we regard bruxism as a symptom and not as an illness in itself, we do not intend to try CBZ or 5-HTP on their own on bruxist Down children, and neither are we in the operative position to be able to do so. We have already cited how in one of our cases 5-HTP on its own was ineffective for at least one year at a dosage of 50 mg daily.

If others would like and are in a position to carry out this type of experiment we would be pleased to learn of the results which will surely help us to understand this symptom further.

As for ourselves, we have promised to re-examine the results of this study in term of analysis of variance, in order to be able to produce, if possible, more thorough and precise indications.

If however, as we maintain, the reactions to stress depend on the constitution of the individual through his/her genetic and acquired components, we may find difficulty, for this very reason, in elaborating standard therapy frameworks.

References

Cocchi R.: Terapia farmacologica nella sindrome di Down: inquadramento teorico. In: Cocchi R., Belacchi C., Cercolani P. (a cura di): Risultati di 8 anni di terapia farmacologica nella sindrome di Down. GISSTIMMAI, Pesaro 1997: 19-41.

Cocchi R., Lamma A.: Bruxism in soggetti affetti da sindrome di Down. Studio epidemiologico su 366 casi. Odontostoinat. Implantoprot. 1987, n.4: 66-69.

Geller M., Kaplan B., Christoff N.: Treatment of dystonic symptoms with carbamazepine. In: Eldridge R., Fahn S. (eds): Advances in neurology, Vol.14. Raven Press, New York, 1976: 404-410.

Isgreen W.P, Fahn S., Barrett R.E., Snider S.R., Chutorian A.M.: Carbamazepine in torsion dystonia. In: Eldridge R., Fahn S. (eds): Advances in neurology, Vol.14. Raven Press, New York, 1976: 411-416.

Morse D.R.: Stress and bruxism. J. Hum. Stress 1982, 9: 34-54

Lamma A.: La patologia dentale nei soggetti Down: il bruxismo. In: Cocchi R., Belacchi C., Cercolani P. (a cura di): Risultati di 9 anni di terapia farmacologica nella sindrome di Down. GISSTIMMAI, Pesaro 1987: 169-180.

Voronina T.A. Pharmacological properties of nicotinamide as a possible ligand of benzodiazepine receptor (Russ). Farmakol. Toksikol. (Moscow) 1981, 44: 680-683.