SIALORRHEA (OR DROOLING)
IN DOWNS. AN EPIDEMIOLOGICAL INVESTIGATION ON 510 Ss.
Renato Cocchi, a
neurologist and a medical psychologist.
Summary.
In a consecutive series of
510 Downs, selected with the exclusion of the psychotic persons, 103 subjects =
20.20 %, have shown sialorrhea during the first examination. This group has
normal M/F ratio , normal distribution of the chromosomal anomalies, average
age to the first examination = 88.67 +/- 65.54 months.
Compared with 300 Ss
with sure sialorrhea absence, both divided in age day bands of a year, till 20
years there is a high ( p <0.0009 ) inverse correlation with the age. One
subject of 25 years with sialorrhea was also found.
The sialorrhea is an
aspecific symptom, without any with the chromosome 21, and probably it is a
symptom of internal metabolic stress with district parasympathetic
overstimulation, coming from the brain..
Key words: Down
Syndrome, sialorrhea, epidemiology, stress.
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Sialorrhea, or excess of
drooling, is a symptom in many persons with many different illnesses such
as:Cerebral palsy; Palsies of the cranial nerves VII, IX, and XII; Autism and
PDD; Mental retardation; Stroke; Amyotrophic lateral sclerosis; Down syndrome;
Motor neuron disease;Parkinsonism; Congenital suprabulbar palsy; Head trauma;
Encephalitis; Heavy metal intoxication; Graves' disease (a hypertiroidism);
Rabies; Extra-esophageal reflux disease; Nasal and/or pharyngeal obstruction;
Major surgical resection of structures in the oral cavity/pharynx/hypopharynx.
It is a symptom
thant can also be induced by drugs, during antipsychotic therapies.
Drooling also impairs
socially the sufferers from it and isolation is unfortunately frequent because
saliva soils furniture, carpets, toys, and the clothing of peers, siblings,
parents, relatives and caregivers.
The saliva production comes
out from both the major and minor salivary glands, the secretory action of
which is mainly under the control of the parasympathetic nervous system.
Excessive drooling is a condition that can induce heavy hygienic and
psychosocial problems.
Although the oversalivation
and drooling in Down syndrome Ss is a well known problem, I did not find any
epidemiological investigation on this symptom. So, following I researched on
Internet on Google, and on Mediline since 1960, by using sialorrhea,
oversalivation, drooling, Down syndrome as key words.
It early attracted my
attention when I started drug therapy in these persons (Cocchi, 1993) and now I
have verified its presence in the clinical cards of 510 Downs.
Materials and methods
This investigation used
the clinical cards referring to a consecutive, series of home reared and home
living Downs as seen in outpatients' clinic by the present author. Psychotic
subjects were excluded .
During their 1st
consultation I wrote the presence of sialorrhea in the referring card.
From all the records I
discarded those pertaining to autistic or PDD Down Ss because I saw that this
second heavier pathology can modify every sympotm or behaviour.
From the remaining
records I collected:
- gender;
- chromosomal
diagnosis;-
- age at 1st
consultation;
- positive or negative
presence of the symptom "sialorrhea" .
I processed data by gender,
chromosomal anomalies, age bands and I applied Chi Square Test, when suitable.
Results
I reported the data
pertaining to the subjects of the clinical records in the following tables.
Tab. 1: Epidemiological data of the
whole series.
|
Nr. of Ss |
510 |
100.00% |
|
Malesi |
292 |
57.25% |
|
Females |
218 |
42.57% |
|
M/F ratio |
133.94/100 |
|
|
|
|
|
|
Chromosomal diagnosis |
|
|
|
Standard trisomy 21 |
461 |
90.39% |
|
Mosaicisms |
16 |
3.14% |
|
Translocations |
16 |
3.14% |
|
Unknown, only clinical diagn. |
17 |
3.33% |
|
|
|
|
|
Age at 1st consultation (months) |
|
|
|
Range |
6-510 |
|
|
Mean +/- SD |
71.37 +/- 69.71 |
|
As we can see in the Table
1, the M/F ratio overlaps the same known ratio of Italian live-born Down
babies. Even the distribution of chromosomal anomalies fitted the variance
range for Italian and International samples.
For these reasons we
ought to maintain the present sample as representative at least of the Italian
Downs population.
Tab. 2. Distribution of the prevalence of
the symptom "sialorrhea."
|
No. of the whole series Ss |
510 |
100.00% |
|
|
||
|
Not investigated (?) |
107 |
20.98% |
|
With sialorrhea (+) |
103 |
20.20% |
|
Without sialorrhea (-) |
300 |
58.82% |
The symptom is present in
20.20% of the whole series, and much probably this is the exact rate, because
almost certainly the not investigated Ss (?) were without it. If they had had sialorrhea
at the first consultation, or at the first checkup, I had surely observed it.
On the other hand, being a symptom that disturbs the parents, they would have
attracted my attention on it, if I missed it.
Tab. 3: Epidemiological data of the sialorrhea
group.
|
|
No. of Ss |
% |
|||
|
Ss with sialorrhea (+) |
103 |
100.00 |
|||
|
Males |
59 |
57.28 |
|||
|
Females |
43 |
42.72 |
|||
|
M/F ratio |
137.21 |
||||
|
|
|||||
|
Chromosomal diagnoses |
|
||||
|
Standard trisomy 21 |
92 |
89.32 |
|||
|
Translocations |
4 |
3.88 |
|||
|
Mosaicisms |
1 |
0.97 |
|||
|
Only clinical diagnosis |
6 |
5.83 |
|||
|
|
|||||
|
Age at first consultation. |
|
||||
|
Average +/- SD (monthe) |
88.67 +/- 65.54 |
||||
|
Range (months) |
6-308 |
||||
The usual male prevalence
is kept up, even if slightly greater of what found at the birth for Italian
children from Camera and Mastroiacovo, 1984.
The distribution of the
chromosomal anomalies parallels what reported for Italian and international
Down populations.
The average age at the
first consultation is higher than that of the whole series, but the age range
is narrower.
It is to observe that a
person with more than 25 years (308 months of age) is a carrier of sialorrhea.
Tab. 4. Epidemiological data of the group
without sialorrhea.
|
|
No. of Ss |
% |
|||
|
Ss without sialorrhea (-) |
300 |
100.00 |
|||
|
Males |
186 |
62.00 |
|||
|
Females |
114 |
38.00 |
|||
|
M/F ratio |
163.16 |
||||
|
|
|||||
|
Chromosomal diagnoses |
|
||||
|
Standard trisomy 21 |
275 |
91.66 |
|||
|
Translocations |
8 |
2.67 |
|||
|
Mosaicisms |
9 |
3.00 |
|||
|
Only clinical diagnosis |
8 |
2.67 |
|||
|
|
|||||
|
Age at first consultation. |
|
||||
|
Average +/- SD (monthe) |
69.36 +/- 70.59 |
||||
|
Range (months) |
6-510 |
||||
At the first eye sight the
M/F ratio increase, while the distribution of chromosomal diagnoses is normal.
The average age at the first visit is lesser than that of the sialorrhea group,
with wider age range.
Tab. 5: Epidemiological data of the group
not investigated for the sialorrhea.
|
|
No. di Ss |
% |
|||
|
Ss not investigated (?) |
107 |
100.00 |
|||
|
Males |
54 |
50.47 |
|||
|
Females |
53 |
49.53 |
|||
|
M/F ratio |
101.87 |
||||
|
|
|||||
|
Chromosomal diagnoses |
|
||||
|
Standard trisomy 21 |
94 |
87.85 |
|||
|
Translocationsi |
4 |
3.74 |
|||
|
Mosaicisms |
6 |
5.61 |
|||
|
Only clinical diagnosis |
3 |
2.80 |
|||
|
|
|||||
|
Age at first consultation. |
|
||||
|
Average +/- SD (monthe) |
65.13 +/- 72.94 |
||||
|
Range (months) |
6-475 |
||||
The group not investigated
Ss has a lower M/F ratio, a distribution of the chromosomal anomalies that differs
a little from the normal distribution. It happens so because a modest deficit
of the standard trisomy 21, and a modest excess of mosaicisms. The average age
at the first visit is the lower, among the three groups.
Tab.6: Comparison of rates for age bands,
between the sialorrhea group and that with sure absence of it.
|
Age band (months) |
With sialorrhea |
% |
Without sialorrhea |
% |
|
1-12 |
5 |
4.85 |
35 |
11.66 |
|
13-24 |
11 |
10.68 |
50 |
16.67 |
|
25-36 |
14 |
13.59 |
42 |
14.00 |
|
37-48 |
8 |
7.77 |
27 |
9.00 |
|
49-60 |
8 |
7.77 |
27 |
9.00 |
|
61-72 |
3 |
2.91 |
16 |
5.33 |
|
73-84 |
5 |
4.85 |
15 |
5.00 |
|
85-96 |
3 |
2.91 |
10 |
3.33 |
|
97-108 |
7 |
6.80 |
8 |
2.67 |
|
109-120 |
9 |
8.74 |
17 |
5.67 |
|
121-132 |
6 |
5.82 |
7 |
2.33 |
|
133-144 |
4 |
3.88 |
14 |
4.67 |
|
145-156 |
5 |
4.85 |
5 |
1.67 |
|
157-168 |
3 |
2.91 |
3 |
1.00 |
|
169-180 |
4 |
3.88 |
4 |
1.33 |
|
181-192 |
3 |
2.91 |
6 |
2.00 |
|
193-204 |
0 |
0.00 |
4 |
1,33 |
|
205-216 |
0 |
0.00 |
0 |
0.00 |
|
217-228 |
0 |
0.00 |
1 |
0.33 |
|
229-240 |
1 |
0.97 |
1 |
0.33 |
|
241+ |
4 |
3.88 |
8 |
2.67 |
|
Totals |
103 |
100.00 |
300 |
100.00 |
Chi Square for the ratesi =
202.589 with 20 df and p < 0.0001
The test shows that the two
groups significanly belong to different populations but it is possible that the
sialorrhea has some link with the age. The presence of the symptom in four
persons older than 20 years not disconfirm such a possibility.
Graph 1
Linear
regression and correlation, till the age of 20 years: no. 20
b
= -0.47; a = 9.70 ; SE b: 0.09; SE a :1.12, estim. SE 2.42 (There is a linear
reverse regression, so cases of doolings decrease with the age )
r
= - 0.761; t = -4.978, con 18 gl e p < 0.0009 (There exists a
significant correlation with the age, as calculated till 20 years, with clear
reduction of persons with sialorrhea).
Discussion.
The sialorrhea is a not specific
symptom that is found in subjects with mental retardation, with or without
genetic defects, in psychotic subjects and other persons. In Italy, in the area
of Pesaro-Ancona there exist families that have their last name as Bavosi (The
Drooling). This fact does think that could exist a some genetic-hereditary
component that has given origin to this last name.
This symptom cannot
directly depend on the chromosomal trisomy 21; Otherwise, all the Down should
have it.
The prevalence found in
this series equals 1 out of 5 Downs (20.20%) and it has a significant
correlation with the age, in the sense that it decreases when the age
increases, at least till the maximum age of the subjects of this series (42
years and half). It is possible however that such correlation becomes less
evident during the senile age (some elderly person started drooling).
I think that sialorrhea is
a symptom of internal metabolic stress, with a district parasympathetic
overstimulation from the brain. If so, and I think it be so, it should modify
following an antistress drug therapy. I shall verify it in the next research on
this topic.
References
Camera G., Mastroiacovo P.:
Epidemiologia della sindrome di Down. In. Ce.Pi.M. (ed): Aspetti
epidemiologici, genetici, clinici, riabilitativi e sociali della sindrome di
Down. Ce.Pi.M., Genova 1984: 225-230
Cocchi R. Drug therapy in
Down's syndrome: A theoretical context . It. J. Intellect. Impair. 1993, 6:
143-154.
Posted on Internet on January 2004.
Copyright by Renato Cocchi 2004
Author's address: dr Renato
COCCHI, via Mercalli 10
42100 Reggio Emilia (Italy)
renatococchi@libero.it
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