Original Article
DOI: http://dx.doi.org/10.1590/1413-785220212904243419
Pediatric Orthopedics
BROAD PECTUS EXCAVATUM TREATMENT:
LONG TERM RESULTS OF A BRAZILIAN TECHNIQUE
TRATAMENTO DO PECTUS EXCAVATUM AMPLO:
RESULTADOS A LONGO PRAZO DE UMA TÉCNICA BRASILEIRA
Davi de Podestá Haje1,2 , Sydney Abrão Haje (in memoriam) 1 , José Batista Volpon3
Ana Carolina Oliveira da Silva2 , Leonardo Ferreira Braz Lima2 , Wilson Huang2
,
1. Centro Clínico Orthopectus, Brasília, DF, Brazil.
2. Hospital de Base do Distrito Federal, Department of Orthopedic Surgery, Brasília, DF, Brazil
3. University of São Paulo, School of Medicine of Ribeirão Preto, Department of Biomechanics, Medicine and Rehabilitation of the Locomotor System, Ribeirão Preto, SP, Brazil.
ABSTRACT
RESUMO
Objective: This study aims the treatment results of broad pectus
excavatum after a long-term follow-up and skeletal maturity. Methods:
Eighty-four children and adolescents with broad-type pectus excavatum
were selected for evaluation after treatment with a dynamic orthosis
that applies compression to the lower rib projections and prescription
of exercises. The broad pectus excavatum was defined as a deformity
that the depressed area was greater and covered the area above and
below the nipple line. All patients were evaluated for more than 1 year
after the end of treatment and skeletal maturity. Post-treatment results
were categorized as mild, moderate and severe. Statistic correlations
between results and deformity flexibility, deformity severity, and adherence to treatment were assessed. Results: The mean age at the
beginning of treatment was 13.3 years, and the follow-up duration
was 25.7 months after suspension of orthosis use. Forty-eight percent
of patients showed good results. With regular use of orthoses and
performance of exercises, this rate increased to 70% (p < 0,001). Mild
cases showed more success than severe cases (p = 0,007). Initial
flexibility didn’t influence the results (p = 0,63). Conclusion: Treatment
of broad pectus excavatum with orthoses and exercises led to good
definitive results in most resilient patients, especially in those with mild
deformities. Level of Evidence V, Expert Opinion.
Objetivo: Estudar os resultados de longo prazo e com seguimento até
a maturidade esquelética do tratamento do pectus excavatum amplo.
Métodos: 84 crianças e adolescentes foram tratados com uma órtese
que aplicacompressão nas saliências costais inferiores, associada a
exercícios específicos. A deformidade foi classificada como ampla
quando a depressão tem maior extensão e abrange uma área acima
e abaixo da linha mamilar. Os resultados foram categorizados em
ruim, regular ou bom, sendo correlacionados estatisticamente com a
flexibilidade, a gravidade da deformidade e a adesão ao tratamento,
com avaliação um ano após o fim do tratamento e na maturidade
esquelética. Resultados: A idade média no início do tratamento
foi de 13,7 anos e o seguimento médio foi de 25.7 meses após a
suspensão do uso da órtese. 48% dos casos apresentaram sucesso
com o tratamento, mas quando os exercícios e o uso da órtese foram
regulares, esta taxa aumentou para 70% (p < 0,001). Os casos mais
leves tiveram maior sucesso que os de maior gravidade (p = 0,007),
mas a flexibilidade inicial não influenciou os resultados (p = 0,63).
Conclusão: O tratamento do pectus excavatum amplo com o uso de
órtese e exercícios apresentou bons resultados definitivos na maioria
dos pacientes resilientes, em especial nos casos mais leves. Nível
de Evidência V, Opinião do Especialista.
Keywords: Braces. Funnel Chest. Pectus Excavatum. Orthotic
Devices. Thoracic Wall.
Descritores: Braquetes. Tórax em Funil. Pectus Excavatum. Aparelhos Ortopédicos. Parede Torácica.
Citation: Haje DP, Haje AS, Volpon JB, Silva ACO, Lima LFB, Huang W. Broad pectus excavatum treatment: long term results of a brazilian technique.
Acta Ortop Bras. [online]. 2021;29(4):197-202. Available from URL: http://www.scielo.br/aob.
INTRODUCTION
In pectus excavatum, we can observe an area of depression
that can be localized or broad, symmetrical or asymmetrical
and may be associated with varying degrees of lower costal
arch protrusions.1-3 The most common reasons that patient or
family members opt for treatment of these deformities are embarrassment and body-image issues.4 Severe pectus excavatum
deformities may be associated with exercise intolerance and
shortness of breath.5
The most widely used treatment for pectus excavatum is surgery.6
Another option is non-invasive treatment with use of a brace and
performance of specific physical exercises. This technique was
initially described by Haje in 1979,7 followed by reports of good
results in a large percentage of patients with pectus carinatum.8,9
All authors declare no potential conflict of interest related to this article.
The study was conducted at Centro Clínico Orthopectus, DF, Brazil
Correspondence: Davi P. Haje. SMHN 2 Bloco A, Ed. Clínicas, sala 804-806, Brasília, DF, Brazil. 70710904. davihaje@yahoo.com.br
Article received on 09/10/2020, approved on 11/05/2020.
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Intuitively, constant pressure on the anomalous protrusion can lead
to bone remodeling and permanent correction of the deformity.
Although much less is known about brace use for the treatment
of pectus excavatum. In this case, the orthosis has a compressive
effect on the protrusion of the last ribs by lowering them, indirectly
causing bone depression elevation with the performance of specific
exercises. This technique was developed by Haje et al.1-3,9,10 who
reported that the application of a standardized treatment protocol
is an important factor for achieving good results, especially in more
flexible cases.1,9,10 But previous Haje’s studies did not describe
the brace and exercise treatment long term results for pectus. It is
important to follow patients with musculoskeletal deformities (like
clubfoot, pectus and others) treated in conservative or surgically
ways until their growth period ends because it is possible to have
relapses and the final result may change.6,11
In addition to this orthosis, reports describes non-surgical treatment
of pectus excavatum using a device called the Vacuum Bell®;12
use of the dynamic chest compressor (DCCII) is associated with
Vacuum Bell.1,13
This study targets the assess long-term results of a non-invasive
method for correction of broad-type pectus excavatum using the
method described by Haje,9 involving a special orthosis for use
with specific physical exercises.
MATERIALS AND METHODS
Patients were treated between 1977 and 2017 according to a pre-established protocol and supervised by one of the authors (DPH or
SAH). Overall, 573 patients with broad-type pectus excavatum were
identified. We excluded 141 untreated patients (105 with non-indications for treatment and 36 patients who did not consent to therapy),
those with follow-up of less than one year (n=125), those with an
iatrogenic etiology (n=2), those who had used a vacuum bell (n=14),
and those who ceased treatment (n=31). Patients who did not attend a
follow-up after 1 year of treatment completion and at skeletal maturity
were also excluded (n=176) because the study objective was not to
verify the immediate or short term results. Only patients who were
followed-up for more than 36 months and 12 months after they had
finished growing were included, totalizing 84 patients.
Pectus excavatum was classified as broad when the deformity
was greater and covered an area above and below the nipple line
(Figure 1).1,2,3 The other form of pectus excavatum is localized and
will be evaluated in another publication.
Figure 1. Broad-type pectus excavatum. The area of deformity is
extensive and often presents with changes to the lower coastal arch
cage which compromises aesthetics.
198
The deformities were classified as mild, moderate, or severe
based on clinical findings and as determined by the physician
(Figure 2). The flexibility of the deformity was evaluated using
a manual compression maneuver of the lower rib cage in the
anteroposterior direction performed by the examiner. Simultaneously, patients were asked to perform the Valsalva maneuver
together with adduction against upper limb resistance. Younger
patients were requested to blow a balloon. The effect of this
maneuver on the excavatum was observed. The deformity was
classified as “flexible” when there was complete reversal of the
depression, “rigid or poorly flexible” when the depression did
not change or changed slightly, and “moderately flexible” with
partial correction (Figure 3).9 Physical examination was performed
only by the first or second author.
A
B
C
Figure 2. Classification of the severity of broad-type pectus excavatum,
according to aesthetic impairment. (A) Mild; (B) Moderate; (C) Severe.
A
B
Figure 3. (A) Flexibility test for pectus excavatum; (B) with compression
of the lower rib cage, in this case performed by the dynamic chest
compressor itself.
Treatment was indicated for moderate or severe deformities in
children and adolescents. Treatment was indicated in some mild
cases when the pectus was noteworthy to the adolescent and
accepted by the family. We indicated treatment before puberty
especially if the deformity started getting worse. Adolescents
with hyperkyphosis were encouraged to start treatment. Patients
with mild scoliosis (less than 20º curvature as determined using the Cobb radiographic method) associated with the pectus
(regardless of its severity) were also encouraged to start treatment.
Only growing individuals with personal and family motivation and
acceptance were treated.
The orthosis is shown in Figure 4. Patients were instructed to wear
the orthosis 23 hours per day (minimum 18 hours). Removal was
allowed during the day for aquatic activities and contact sports.
The compression force was controlled by the patient and maintained
at a comfortable level. Progressive compressive adjustment was
interrupted when the costal salience disappeared. Along with
the use of the orthosis, specific exercises were recommended to
strengthen the muscles of the anterior chest wall. Weaning from the
orthosis was performed gradually until the minimum usage period
of 24 months was completed.
Acta Ortop Bras. 2021;29(4):197-202
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categories of a third variable. The level of significance was p ≤ 0.05.
The Statistical Package for Social Sciences (SPSS) v.22.0 (IBM Corp.,
Armonk, NY, USA) was used for all statistical analyses.
RESULTS
Figure 4. Type of brace used for the treatment of broad-type pectus
excavatum. The two anterior cushions exert pressure on the last costal arches and are connected to a posterior cushion by means of a
threaded bar that serves to control the compression force.
The mean age at the beginning of treatment was 13.3 years (standard
deviation [SD] = 3.5 years; median = 13,0 years, variation 4,8-17,8),
being 19% (n = 16) less than 10 years old. The age histogram of the
84 treated patients that completed the follow-up is shown in Figure 5.
Of the treated patients, 66 (79%) were male and 18 (21%) were female.
The mean follow-up duration was 25.7 months after the end of treatment.
20
Statistical analysis
The chi-square test (χ2) was used in the analysis of categorical
variables and also Cochran-Mantel-Haenszeltest to determine whether the odds ratios between two variables remained the same for
A
15
Frequency
Adherence to treatment was classified as irregular when the orthosis
was not used for the prescribed time, or no regular performance of the
physical exercises. Patients who ceased orthosis use before discharge
were excluded; but those who only ceased performing the exercises
before the recommended period were classified as showing irregular
adherence. Results were considered poor when the deformity did not
change or worsened, average when there was under-correction, and
good when there was significant improvement. The treatment results
were analysed by either the first or second author of this manuscript.
Treatment satisfaction was reported by the family (when the patient was
a child) or by the adolescent patient as “satisfied” or “not satisfied”.
Treatment success was defined as a good result and if the patient was
satisfied. All patients were photographed in the same positions, before
and after treatment, and clinical images were used in the evaluation.
The comparative group consisted of individuals who met indication
for treatment but refused and then returned for re-evaluation.
The evaluation protocol was approved by our Institutional Ethics
Committee (58417516.3.0000.5553).
10
5
0
0.00
5.00
10.00
15.00
20.00
Age
Figure 5. The histogram of age of the treated patients that completed
the follow-up.
Eleven patients (13%) presented with mild initial deformity, 55 (66%)
were moderate, and 18 (21%) were severe. The deformities were
very flexible or moderately flexible in 50 patients (59 %) and were
rigid or not very flexible in 34 patients (41%).
Results were considered good in 40 patients (48%), average in
36 patients (43%), and poor in eight patients (9%). Figures 6 and
7 show a good result. Treatment satisfaction was reported in all
patients with good results, in 20% (n = 17) of those with average
results and in none with poor results.
B
C
Figure 6. A patient aged 15 years and 6 months at the beginning of treatment (A), with good results obtained less than 12 months after the
beginning of treatment and maintained improvements after 28 months of follow-up (B), with maintenance of correction at 23 years-of-age, with
9 years of follow-up, and 5 years after complete interruption of treatment (C).
A
B
C
D
E
F
Figure 7. (A) A patient aged 13 years and 6 months showing pectus excavatum and hyperkyphosis (B_ with natural posture; (C) and with posture corrected by the examiner, (D) with a good result after 3 months of treatment, which was maintained regularly until 15 years and 6 months of age, when
orthoses weaning started, (E) with results maintained after 6 years of follow-up, and after 3 years of treatment interruption. He presented with correction of
hyperkyphosis and protrusion of the lower ribs (see improvement of the positioning of the red line before treatment in B and C, and at treatment end in F).
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Good results were achieved in 72% (n = 8) with mild deformities,
47% (n = 26) with moderate deformities, and 33% (n = 6) with
severe deformities. Mild cases showed more success than severe
cases (p = 0,007).
Regarding flexibility, the results were good in 48% (n = 24) with
flexible or moderately flexible deformities and 47% (n = 16) with
rigid or less flexible deformities.
The use of brace was regular in 69% (n = 58) and irregular
in 31% (n = 26). Overall, 54% (n = 45) of patients performed
the specific exercises regularly, while 46% (n = 39) performed
these irregularly. When flexibility was analyzed in isolation,
no significant differences is shown among the various results
(p = 0,63). Even when flexibility was analyzed together with
the regularity of the use of the orthosis and performance of the
exercises, there was no significant influence on the response
to treatment (p = 0,67).
Seventy percent of patients with regular orthosis use and
exercise performance achieved good results (p < 0,001). By
contrast, when orthosis use was irregular, regardless of the
regular performance of the exercises, only 29% of patients
achieved good results (p = 0,84). Table 1 shows the associations
between variables.
Table 1. Comparisons made and interpretation of results
Comparison
χ2 value / P /
odds ratio
and C.I.95%
Association
between variables
Good results vs.
pectus severity
16,3 / p = 0,007 /
3,8 (1,1-13,4)
Negative
Good results vs.
pectus flexibility
0,23 / p = 0,63 /
0,84 (0,42-1,7)
No correlation
Good results vs. regular
orthotic wearing
24,68 / p < 0,001/
10,65 (1,74-17,9)
Positive
Good results vs. regular
physical exercises
8,57 / p = 0,04 /
4,36 (2,4-8,89)
Positive
Good results vs. regular
orthotic wearing and
regular physical exercises
15,68 / p < 0,001 /
5,22 (2,15-12,64)
Positive
Good results vs. irregular
brace wearing and regular
physical exercises
3,35 / p = 0,84 /
1,22 (0,38-3,27)
No correlation
No major complications were observed; but skin irritation or transient
hyperpigmentation occurred in the areas of pressure in about 4% of
patients. In 12% of patients, was identified discomfort or transient
pain in the orthosis support areas, both of which resolved with
partial release of pressure. These complications did not lead to
an interruption in orthosis use. Relapses in those patients that
completed the follow-up were not showed.
Thirty-six patients or family members did not accept treatment;
however, nine returned to be re-evaluated. Seven of these (78%)
presented worsening of the deformity (Figure 8) and two were
stable. All patients with worsening of deformity presented with
moderate or marked deformities, with four (57%) defined as
moderately flexible and three (43%) defined as rigid at the last
evaluation (approximately 15.2 years of age). Some treated patients
who presented with some improvement returned with recurrence
of the deformity when they stopped the treatment before medical
orientation (n = 5) (Figure 9).
200
A
B
Figure 8. (A) A 10-year-old boy with moderate deformity, where the
family did not accept the indication for treatment. (B) At 14 years-ofage, the deformity is worse (B).
A
B
C
D
Figure 9. (A)The patient started treatment at 12 years of age; (B) with
good results after 12 months of regular orthosis use and irregular
exercise adherence. (C) Besides, the patient abandoned treatment.
In adolescence, the deformity recurred, and treatment was restarted.
(D) After 1 year, there was some improvement.
In two patients, a slight hypercorrection (depressed area turned into
a pectus carinatum) was presented and managed by reducing the
orthosis DCC II usage time or the use of a second orthosis for the
treatment of pectus carinatum, called DCC I (Figure 10).
A
B
C
Figure 10. (A) An 11-year-old boy with moderate excavatum deformity
who presented with hypercorrection; (B) after 24 months of treatment
with development of mild pectus carinatum; (C) that was controlled
using another orthosis.
DISCUSSION
In terms of treatment, surgical correction of pectus is considered
by most surgeons as the only effective option. The aesthetic
results of surgery are not always the desired ones; for example,
the possibility of maintaining prominent costal edges and the
possible appearance of a reactive pectus carinatum is accepted.14
Still, surgery poses serious complications, and life-threatening
complications are often underestimated, including subsequent
removal of the implant.15 This makes the decision to proceed with
surgical treatment very difficult for some patients, especially those
with mild or moderate pectus.
When choosing initial pectus treatment for growing patients, we recommend first considering non-invasive treatment methods such as
those based on orthosis use (bracing) and physical exercises,as
a low-risk method with the possibility of interruption in cases of
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intolerance or change of opinion is possible. Failure to recommend
treatment in cases of skeletally immature pectus excavatum may
constitute negligence because the patient’s condition may worsen
during their growth and because milder deformities showed better.
In our opinion, surgery should be indicated for patients who cannot
adapt or did not show good results with the available conservative
therapy, as well as those who have severe deformities and psychological issues. Finally, the patient and their family must accept
the risks imposed by surgical treatment.
48% of the patients, had good results, suggesting that the broad
form of pectus excavatum can be efficiently corrected using
the non-invasive method described here as a group of patients
followed skeletal maturity. Haje and Haje (2009),3 who treated
localized and broad pectus excavatum with a brace and exercises, reported good results in 29% and 21% of patients after a
follow-up of less than 1 year, respectively. Moon et al.16 reported
that their results were long-term; despite, they accepted a minimum
follow-up of only 13 months in cases of pectus carinatum treated
with orthosis; we believe that a longer observation period to growth
stabilization and an additional observation period after finishing
treatment are essential.
In pectus excavatum, the pressure exerted by the orthosis on the
lower anterior costal arches associated with exercises probably
increased the pressure in the mediastinum with consequent expansion of the osteo-cartilaginous structures of the chest’s anterior
face, modifying the forces that act on the various growth plates
and contributing to the remodeling of deformed structures. Wolff’s
law17 explains the remodeling process of skeletal structures. Wong
and Carter reported that mechanical forces on the sternum may
influence skeletal morphogenesis.18 In PE (pectus excavatum)
patients, who reportedly have increased midline excursion at the
umbilicus level,19 the braces and specific exercises might help limit
that excursion. The action of the diaphragm may become more
efficient to expand the chest as the protrusions of the costal edges
are corrected by presence of the orthosis. The performance of
specific and repetitive exercises associated with orthosis use, along
with holding the maximum inspiration during muscle contraction,
works actively to correct the depressed area of the pectus and
stimulates postural improvement.
This study highlights some prognostic factors that are very useful
during the initial treatment period. A positive correlation between
good results and treatment adherence is observed, including
regular orthosis use and regular performance of the associated
exercises. We believe that pectus excavatum treatment with the use
of orthoses and exercises should begin fast because we found that
more severe deformities were related to worse results. Despite that,
the correlation between flexibility and good results is not showed.
Haje et al.1,3 found that flexibility was an important prognostic factor
in the treatment with orthoses and specific exercises.
The method used in this study had a low rate of complications.
The most frequent complication was skin irritation, which did not
lead to treatment interruption.1,3 The other rare complication, hypercorrection,10 was very well managed with treatment adjustment
and use of an orthosis used for reactive pectus carinatum.
Some limitations were shown in this study, most of which were
based on subjective criteria used to define pectus type, its severity and flexibility. This limitation could be minimized with image
evaluation methods that do not involve exposure to radiation, such
as 3D structured light scanners, 20 but this exam was not currently
available The authors were concerned about using a CT before
and after treatment because of radiation, costs and that would
not change the braces and treatment indication criteria. Besides,
the description of an objective flexibility test for pectus excavatum
was not identified. The authors think that analyze results using
photos in the same position before and after treatment is enough.
The fact that all patients classified as a good result by the authors
had satisfaction with their final result, brings more credibility to
the treatment results.
CONCLUSION
In conclusion, we found that the non-invasive method for broadtype pectus excavatum correction presents good results in the
patients followed until skeletal maturity and those who adhered to
the treatment protocol, although it is a long treatment process that
may lead to short or late loss of follow-up, treatment irregularity,
or abandonment of treatment before the deformity is stabilized.
AUTHORS’ CONTRIBUTIONS: Each author contributed individually and significantly to the development of this article. DPH: conceived and planned the
activities that led to the study, participated in the review process, performed the treatment together with Sydney Haje from 1999 to July 22th 2011, and by
himself since this last date, interpreted the results of the study and approved the final version; SAH (in memoriam): Performed the treatment from 1988 to
July 22th 2011 (3 days before he passed away); JBV manuscript preparation, participated in the review process and approved the final version; ACOS: data
collection and approved the final version; LFBL: data collection and approved the final version; WH: data collection and approved the final version.
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