Keywords
sleep-disordered breathing - quality of life - tonsillectomy - adenoidectomy - OSA-18
- child
Introduction
Sleep-disordered breathing (SDB) affects from 7 to 17% of the pediatric population,
and the symptoms range from primary snoring to obstructive sleep apnea (OSA).[1]
[2] The nocturnal symptoms related to SDB are snoring, witnessed apnea, sialorrhea,
restless sleep, and enuresis. The daytime symptoms are hyperactivity, attention deficit
disorder, daytime naps, excessive daytime sleepiness, behavioral disorders (including
aggressivity, anxiety, and depression). Physical impact is also described, including
muscular and skeletal changes. Untreated disease is believed to affect emotion, cognitive
function, and neurobehavior of both children and family members that may contribute
to a poor quality of life (QoL).[2]
[3]
Treatment of SDB is associated with an increase in health care utilization and cost.
Children with SDB, as compared with controls, have a significantly higher rate of
antibiotic use, 40% more hospital visits, and an overall elevation of 215% in health
care usage, mostly from increased respiratory tract infections.[4]
[5]
[6]
[7] Failure to thrive is reported in 27 to 62% of pediatric OSA cases, and up to 40%
of children with SDB exhibit behavioral problems.[8]
Though SDB is a common condition with a high impact on physical, developmental, cognitive,
and behavior aspects in childhood, data from pediatrician knowledge, training and
abilities on treating children with SDB shows there is a lack of knowledge and misinformation
about this subject.[9] One study that evaluated 112 pediatricians demonstrated that the majority of them
(62%) consider that information about SDB during medical school and residency are
scarce, albeit children with SDB are frequently attended at the pediatric office.[10]
Adenotonsillectomy (AT) is the first line of treatment for OSA secondary to adenotonsillar
hypertrophy in children.[2] Data, in 1993, from the National Hospital Discharge Survey noted a decrease of 0.50%
in inpatient tonsillectomy rates from 1977 to 1989,[11] and similar reports from 1978 to 1986 showed that the rate of tonsillectomy for
treatment of throat infections declined; however, the frequency of SDB as the primary
indication for the procedure increased, especially in children under 3 years of age.[12] Although the current indication for ATis well established in literature, misconceptions
about the surgery concerning to risks, benefits and impact on the immunological system
are still present and lead some pediatricians to discourage the caregivers to accept
AT. This is a secure surgery, and published data reported mortality rates for tonsillectomy
from 1 per 2,360 (in inpatient settings) to 1 per 56,000 patients.[13]
[14]
[15] Previous studies show there would, therefore, appear to be a therapeutic advantage
to removing recurrently diseased tonsils because they are no longer able to function
adequately in local protection. However, some studies demonstrate minor alterations
of immunoglobulin concentrations in the serum and adjacent tissues following tonsillectomy.[16]
[17] Nevertheless, there are no studies to date that demonstrate a significant clinical
negative impact of tonsillectomy on the immune system.
The gold standard for the diagnosis of OSA is a polysomnography (PSG). However, PSG
is expensive, requires time and a trained professional, and is often unavailable at
many institutions where children with SDB are treated.[2]
[18] Therefore, most cases of SDB are treated based on clinical assessments and physical
exams, with no objective testing. Albeit it may not be an ideal situation, children
without comorbities may the treated without PSG.[19]
[20] Though PSG is ideal for evaluating the presence and severity of apneas, it fails
to measure the impact of SDB on children's QoL in general, as well as in terms of
its effect on behavior and emotion.[19]
[21] Because of this, the use of questionnaires to evaluate QoL has become popular in
recent years.[22]
[23]
[24]
Recent articles have evaluated the impact of surgery on QoL among children with SDB
through questionnaires on physical, emotional, behavioral, and familial factors.[3]
[21]
[25]
[26]
[27] There are different methods for evaluating postadenotonsillectomy QoL among children,
and the obstructive sleep apnea questionnaire (OSA-18) is one of the most frequently
used questionnaires worldwide. It has been validated in a variety of languages.[28]
[29]
[30]
[31] Most of the studies evaluated children for 6 months or less and rarely the patients
were reassessed over 1 year after AT.[19]
[32]
[33]
[34]
Objective
The present study aimed to describe the QoL in children with SDB and the long-term
impact of AT on this disease.
Method
This was a prospective, non-controlled study that was approved by the research ethics
committee of the institution (decision no. 1.429.954).
This study included patients between 3 and 13 years of age, of both genders, in ASA
classes 1 and 2 who were treated in the otorhinolaryngology department between January
2017 and January 2018 and whose symptoms included snoring, witnessed apneas, noisy
breathing, mouth breathing, and restless sleep. All of the patients were treated by
the same pediatric otolaryngologist. Patients with symptoms consistent with rhinitis
were treated using nasal corticosteroids and/or antihistamines, as recommended for
3 months, and were then reevaluated.[35] Once the child presented complete recovery of the symptoms after clinical treatment,
surgery was discarded. Tonsil size was evaluated using the Brodsky grading scale.[36] Adenoid size (in percentage of obstruction) was calculated through the skull lateral-view
X-ray.[37]
The children were weighed and measured by the principal investigator. Patients with
overweight, obesity, underweight, craniofacial malformations, genetic syndromes, neuromuscular
diseases, or developmental delays were excluded, as were patients who required a concomitant
procedure (including inferior turbinate surgery).
The patients selected for surgery were informed of the study and were officially included
once the guardians signed the informed consent form, and the children signed the informed
consent form when possible. The OSA-18 was administered by the hospital social worker
1 week before, and then again 10 days, 6 months, and 12 months after the procedure;
another assessment was done in July 2019. The first two assessments were in person,
and the other evaluations were made by phone.
The OSA-18 is a validated questionnaire that contains 18 questions divided into 5
domains: sleep disturbance, physical suffering, emotional suffering, daytime problems,
and caregiver concerns. Each domain receives a score from 1 to 7; higher scores reflect
a worse QoL. Total scores lower than 60 suggest that the sleep disorder has little
impact on QoL; scores between 60 and 80 indicate a moderate impact, and scores above
80 reflect a high negative impact of the sleep disorder on QoL.
The statistical analyses were performed using the IBM SPSS software version 22.0 (IBM
Corp., Armond, NY, USA). The normality of the samples was evaluating using the Kolmogorov-Smirnov
test, which allowed for the use of parametric tests in the data analysis. The Chi-squared
test and analysis of variance (ANOVA) were used to evaluate the data, and p-values lower than 0.05 were considered statistically significant.
Results
Fifty-one patients were evaluated, out of whom 8 were excluded because they presented
complete recovery of the symptoms after treating rhinitis. From the 43 children initially
included in the study, only 31 completed all phases of research. The average age was
5.1 ± 2.9 years, and 16 (51%) of the patients were male. The demographic data and
clinical characteristics of the patients are described in [Table 1].
Table 1
Demographic data and clinical characteristics of the patients
|
Age in years (mean ± SD)
|
5.2 ± 2.9
|
|
Male
|
51%
|
|
Height in cm (mean ± SD)
|
119 ± 18
|
|
Weight in kg (mean ± SD)
|
18.4 ± 7
|
|
Adenoid size (%)
|
79.1 ± 9
|
|
Presence of rhinitis symptoms (n/%)
|
7/ 22%
|
|
Tonsil size (Brodsky grade)
|
Grade 2—17%
|
|
Grade 3—66%
|
|
Grade 4—17%
|
Abbreviation: SD, standard deviation.
The most common clinical findings were snoring (which was an inclusion criterion)
followed by restless sleep, chocking noise, frequent awakening, sialorrhea, witnessed
apnea, and enuresis, as shown in [Table 2].
Table 2
Distribution of sleep-disordered breathing symptoms
|
Symptoms
|
Frequency (%)
|
|
Snoring
|
100%
|
|
Restless sleep
|
92%
|
|
Choking
|
80%
|
|
Frequent awakening
|
73%
|
|
Sialorrhea
|
65%
|
|
witnessed apnea
|
9%
|
|
Enuresis
|
6%
|
|
Hyperactivity
|
3%
|
|
Poor school performance
|
3%
|
|
Aggressive behavior
|
0%
|
The mean preoperative OSA-18 scores were 79.9 ± 12. The scores of all the evaluated
patients improved after surgery (p < 0.05), with mean reduction of 40 ± 8 in the first postoperative evaluation (10
days after surgery). Mean total OSA-18 score after surgery remained under 37 in all
assessments. The last evaluation, done in 2018 July, was done 22 ± 3 months after
surgery (see [Table 3]). None of the patients presented increased values of OSA-18 scores after surgery,
neither in total scores nor in each domain separately.
Table 3
Scores of the obstructive sleep apnea questionnaire before and after surgery
|
OSA-18 scores
|
Mean ± SD
|
p-value[*]
|
|
Sleep disturbance
|
Before
|
16.3 ± 5
|
< 0.001
|
|
10 days
|
7.9 ± 4
|
|
6 months
|
8.0 ± 3
|
|
12 months
|
6.9 ± 3
|
|
Late follow-up
|
5.7 ± 2
|
|
Physical suffering
|
Before
|
21.6 ± 3
|
< 0.001
|
|
10 days
|
8.9 ± 4
|
|
6 months
|
9.0 ± 5
|
|
12 months
|
8.5 ± 4
|
|
Late follow-up
|
8.3 ± 3
|
|
Emotional suffering
|
Before
|
11.5 ± 6
|
< 0.001
|
|
10 days
|
4.1 ± 2
|
|
6 months
|
3.9 ± 2
|
|
12 months
|
4.0 ± 2
|
|
Late follow-up
|
5.0 ± 1
|
|
Daytime problems
|
Before
|
9.2 ± 4
|
0.007
|
|
10 days
|
6.0 ± 3
|
|
6 months
|
6.9 ± 4
|
|
12 months
|
6.6 ± 4
|
|
Late follow-up
|
6.5 ± 4
|
|
Caregiver concerns
|
Before
|
21.8 ± 5
|
< 0.001
|
|
10 days
|
9.0 ± 3
|
|
6 months
|
8.1 ± 4
|
|
12 months
|
7.6 ± 4
|
|
Late follow-up
|
7.2 ± 4
|
|
Total
|
Before
|
79.9 ± 12
|
< 0.001
|
|
10 days
|
36.8 ± 13
|
|
6 months
|
35.3 ± 14
|
|
12 months
|
34 ± 13
|
|
Late follow-up
|
32.9 ± 14
|
Abbreviation: OSA-18, obstructive sleep apnea questionnaire; SD, standard deviation.
* ANOVA.
The reduction in the OSA-18 scores was not different between one domain in relation
to another (p > 0.05). Both male and female experienced improvement of OSA scores before and after
the surgery, without gender effect ([Fig. 1]).
Fig. 1 Obstructive sleep apnea (OSA-18) questionnaire scores before and after surgery regarding
to gender. Analysis of variance. P-value = 0.55.
Tonsil size and adenoid size were correlated with OSA-18 scores before surgery. No
correlation was found between questionnaire scores and the degree of tonsil size,
as detailed in [Table 4]. A correlation was found between adenoid size and OSA-18 scores (p = 0.02), but it was a weak correlation that may be discharged.
Table 4
Correlation between tonsil and adenoid size and total obstructive sleep apnea questionnaire
score
|
Tonsil size
|
Corr (r)
|
17.2%
|
|
p-value[*]
|
0.95
|
|
Adenoid size
|
Corr (r)
|
28.1%
|
|
p-value[*]
|
0.02
|
* Pearson correlation.
Discussion
The present study had described the characteristic of pediatric SDB and long-term
impact of AT on the QoL in an adequate and objective manner.
Preoperatively, only a small percentage of patients presented with symptoms related
to emotional, behavior, and diurnal complaints. All participants were referred for
snoring, and the majority of them for nocturnal symptoms. Previous studies had demonstrated
that snoring and sleep disturbances are the hallmarks for pediatric SDB.[12] Only a small portion of pediatric SDB presented with emotional and diurnal disorders.[3] This characteristic is recognized as one of the significant differences between
adult and pediatric SDB.[3]
[38]
Sleep-disordered breathing can negatively affect children's memory and attention levels,
likely because of hypoxia, which is also considered to be responsible for daytime
symptoms such as irritability, aggression, and hyperactivity.[39]
[40] Furthermore, SDB is associated with enuresis, metabolic problems, and with an increased
risk of metabolic and cardiovascular abnormalities, decreasing children's QoL.[18]
[41]
It is important to evaluate children's QoL because it reflects individual sentiments
toward physical, emotional, and social factors in their life and how these factors
relate to their health.[3]
[21] The OSA-18 is an evaluative, discriminative, and validated instrument to assess
QoL in children with SDB, and it is the most widely used evaluation tool worldwide.[19]
[25]
[31]
An analysis of the literature revealed that AT is the treatment of choice for children
with SDB caused by adenotonsillar hypertrophy. As a result, most of the studies that
evaluate QoL in this group of patients do not have a control group.[31]
[32]
[41] Studies that compare the surgery to watchful waiting have shown that children who
undergo AT exhibit a significant improvement in QoL when compared with children in
the watchful waiting control group.[7]
[42] Our study found a significant improvement in all of the domains assessed by the
OSA-18, similarly to the results found in other studies.[23]
[32]
[43] This improvement can be measured not only by the numerical decrease in the scores
for each domain of the OSA-18, but also through the changes from total scores reflecting
high or moderate impacts on QoL (scores above 60) to total scores reflecting low impact
on QoL, which occurred in the current study and in prior research.[7]
[38]
[44] Our finding that this outcome is maintained for at least 6 months is consistent
with other reports in the literature.[31]
[34]
[42]
[45] Long-term follow-up studies after AT can be difficult to be performed, since most
patients do not need further medical evaluation after improvement of the symptoms
is observed. This is especially noticeable in children without comorbidities, since
the surgical success rate in this group is high. Only 2 studies that evaluated QoL
evaluated children for more than 1 year after AT, and one of them was prospective.
The present study followed the children for 24 months after the procedure.[33]
[46] This is the largest prospective study that evaluated the long-term effects of the
surgery on the QoL of children with SDB using the OSA-18.
Some studies describe reappearance of symptoms during the follow-up, mainly in females
with normal weight and older than 6 years, which was attributed to an incomplete therapeutic
response and due to a change the parental perception of the symptoms.[32]
[44] In our study, none of the patients presented worsening of the OSA-18 values after
surgery. Although allergic rhinitis is considered a risk factor for AT failure, it
was not found in our study.
Tonsil and adenoid sizes are a reason for concern not only for caregivers, but also
for many physicians who are involved in these patients' care. Some studies have found
that there is an association between tonsil size and OSA as well as between tonsil
size and PSG findings, but most of these studies have low levels of evidence.[47] Well-designed studies have shown, as our study found, that tonsil and adenoid size
is not correlated with either SDB severity or patient QoL.[19]
[38]
[47]
[48]
Our study did not find differences between OSA-18 scores and gender, which is similar
to the findings of other studies. The analysis of previous studies that found a higher
prevalence of OSA in males shows that it is probably influenced by age, once this
difference was found only in older boys. It could be explained by hormonal changes
due to puberty, because OSA is more prevalent in male adults.[49]
[50] Since our study group was limited to 13-year-old patients, they probably were not
under hormonal effects yet.
One advantage of our study is that all steps were performed by a single team during
a long-term follow-up. But there are some limitations, including the fact that many
caregivers believe that surgery is the best treatment for the child, particularly
after agreeing to surgery under general anesthesia. This belief may influence caregivers'
answers in the postoperative period.[32]
[51] Another limitation of this study is the lack of the preoperative PSG. This exam
is the gold standard in evaluating children with SDB and is particularly useful for
children without comorbities. However, the difficulty in implementing this exam on
a large scale leads to use other exams or clinical evaluation alone to diagnose and
treat these patients.[30]
[38]
[52] Finally, considering that SDB is a prevalent condition, a large, diverse subject
population must be surveyed to determine whether AT leads to improvement in the QoL
of children,.
Conclusion
This study concludes that SDB in children leads to a poor QoL and that AT improvement
remains for at least 22 months after the surgery. The positive impact of surgery occurs
both in nocturnal and daytime symptoms. Since pediatricians are the medical professionals
with the greatest contact with children with SDB, their knowledge of the subject is
fundamental to these patients' care.
