Idiopathic scoliosis with recurrent dyspnea and wheezing in an otherwise healthy female adolescent: a case report
Article information
Abstract
A 15-year-old female visited our hospital’s outpatient and emergency department several times due to recurrent paroxysmal dyspnea for 6 months. Based on the clinical symptoms, the researchers diagnosed the patient with bronchial asthma and treated her with oral steroids and inhaled corticosteroid agents, but her patients did not significantly improve. At that time, thoracic scoliosis was observed in previous chest X-ray images. Previous records showed that the scoliosis had progressed during the rapid growth period in 2018 (patient age 10 years) and 2023 (patient age 15 years). As the symptoms did not improve despite medication, a further evaluation was performed. Contrast-enhanced chest computed tomography revealed the T8 vertebral body compressing the right bronchus intermedius. The patient rarely wore an existing brace due to discomfort, but a new custom brace was prescribed after confirmation using chest computed tomography. Since then, the patient's compliance with treatment has improved, and she has gradually increased the amount of time she wears the brace. In addition, her Cobb’s angle and pulmonary function tests have improved in outpatient follow-up. The researchers initially considered the patient's recurrent dyspnea and wheezing to be an asthma exacerbation and treated her with medication, but the symptoms did not improve. Therefore, when a patient does not respond well to asthma medications, the possibility of dyspnea or wheezing due to causes other than asthma should not be ruled out. Evaluation for scoliosis should also not be neglected, especially in growing adolescents.
Introduction
Scoliosis is caused by a three-dimensional rotational deformity of the spine [1], and can lead to various forms of respiratory disease and decreased lung function. Idiopathic scoliosis accounts for more than 85% of cases [2], and although the exact etiology is not known, it is presumed that multiple factors are involved. Severe scoliosis requiring surgical treatment is defined as a Cobb angle greater than 45° or more [3]. In general, restrictive lung diseases are pulmonary disorders characterized by a reduced distensibility of the lungs, compromising lung expansion, and reduced lung volumes [4]. However, it can also occur in the form of obstructive lung disease, where the inner diameter of the bronchi is narrowed due to compression. Unlike this, bronchial asthma is a chronic inflammatory airway disease characterized by airway hyperresponsiveness and reversible airway obstruction [5]. This case is a study of a patient who presented with an exacerbation of asthma that did not respond to medication but improved after wearing a brace.
Case
Ethical statements: This study was approved by the Institutional Review Board (IRB) of Busan St. Mary's Hospital (IRB No: 24-14000-134). Written informed consent was waived.
A 15-year-old female visited our hospital’s outpatient and emergency department several times due to recurrent paroxysmal dyspnea for 6 months. This patient was a healthy woman who had a normal vaginal delivery at 38+1 weeks, weighing 3.20 kg, and had no underlying diseases or medical history other than a thyroid nodule discovered incidentally in May 2022. She also had no history or family history of allergic diseases. On initial examination, diffuse polyphonic wheezing auscultated over both lung fields, and decreased aeration was also noted. But a chest X-ray (CXR) shows no increased opacities or consolidation/hyperinflation. Based on the clinical symptoms, the researchers diagnosed bronchial asthma and treated the patient with short-acing beta agonist, short-acting muscarinic antagonist, leukotriene receptor antagonist, oral steroids, and inhaled corticosteroid agents (Table 1), but she did not show significant improvement in her symptoms. Eosinophil, serum immunoglobulin E, hemoglobin, O2 saturation, ImmunoCAP (inhalent), gas analysis values, and transthoracic echocardiogram results were normal at presentation (Tables 2, 3).
At that time, thoracic scoliosis corresponding to Cobb's angle of 70° was observed on CXR. Previous records showed that the scoliosis progressed during the rapid growth period in 2018 (patient age 10 years, 24.1°, Fig. 1A), 2019 (patient age 11 years, 48.3°, Fig. 1B), 2021 (patient age 13 years, 56.6°), and 2023 (patient age 15 years, 70.0°, Fig. 1C). As the symptoms did not improve despite continuous medication, a contrast-enhanced chest computed tomography (CT) was performed, which revealed a thoracic 8 vertebral body compressing the right bronchus intermedius and increased shading of the right middle lobe/right lower lobe (Fig. 2). There were no specific findings in the lung parenchyma on the chest CT. Follow-up CT was not performed because the patient did not want it.
Cobb's angle on chest X-ray. (A) At the patient's age of 10 years (24.1°). (B) At the patient's age of 11 years (48.3°). (C) At the patient's age of 15 years (70.0°). (D) At the patient's age of 15 years (60.2°).
Compression of the right bronchus intermedius by 8th thoracic vertebra confirmed on contrast-enhanced chest computed tomography. (A) Horizontal plane. (B) Coronal plane.
The patient rarely wore the existing brace due to discomfort, but a new custom brace was prescribed after confirmation of the chest CT. Since then, the patient's compliance with treatment has increased and she has gradually increased the amount of time she wears the brace. In addition, her Cobb’s angle has decreased from 70.0° to 60.2° at outpatient follow-up, and her forced expiratory volume in 1 second (FEV1) on pulmonary function tests (PFTs) has increased from 35% to 49% (Table 4 and Fig. 1C, 1D), and she has no significant limitations in her daily life. The improvement in symptoms was indirectly estimated from the significant decrease in the number of visits due to acute dyspnea.
Discussion
In this case, the researchers considered the patient's recurrent dyspnea and wheezing to be an asthma exacerbation and treated her with medication, but the symptoms did not improve. Therefore, when a patient does not respond well to asthma medications, the possibility of dyspnea or wheezing due to causes other than asthma should not be ruled out. Evaluation for scoliosis should also not be neglected, especially in growing adolescents.
This patient was a healthy middle school student with no underlying disease or medical history, and there was no evidence to suggest other causes, such as interstitial lung disease, amyotrophic lateral sclerosis, myasthenia gravis, or ankylosing spondylitis other than idiopathic scoliosis.
Over the past few years, as the COVID pandemic has passed, obesity in children and adolescents has increased due to restrictions on outdoor activities [6], and several studies have shown the relationship between obesity and scoliosis [7] and the relationship between reduced activity and scoliosis [8]. Therefore, when scoliosis is suspected during pediatric treatment, it can be said that screening roles such as standing spine X-ray examination or consultation with other departments are important.
Treatment of scoliosis includes observation, non-surgical treatment, spinal braces, surgical correction and stent treatment depending on the degree of curvature. Non-surgical treatments include physical therapy, superficial electrical stimulation, and chiropractic, but evidence on treatment effectiveness is still lacking [9]. Braces have an initial corrective effect, but since the recommended daily wear time is more than 18–20 hours [9], it is realistically difficult for patients to combine group living and treatment. In fact, a study that followed up on the effect of brace treatment for 8 months showed that the permanent improvement in lung function was limited [10]. Surgery is recommended when the curve of more than 45° is expected to progress further as it grows, or when pain persists even after growth is complete. However, even if the curvature is more than 50°, there is controversy about the necessity of surgery if the patient has completed growth or has no accompanying symptoms [9]. The biggest complication of surgery is infection [9]. According to another study, there was a case where pulmonary function temporarily improved after surgical treatment of scoliosis, but the long-term effect was still questionable [10]. There was also a case report in which pulmonary function was completely restored through stent insertion using a bronchoscope [11].
In this case, brace treatment was selected, and after 4 months, FEV1 and forced vital capacity (FVC) increased on PFTs, but FEV1/FVC ratio decreased. In other words, the increase in FVC was greater than FEV1, which can be interpreted that brace treatment was more effective for restrictive lung disease. The main opinion in the academic world is that there is little therapeutic benefit from braces at the age when growth is generally completed [1]. However, the decline in respiratory ability is not simply related to the angle of scoliosis, but is affected by a number of factors. Therefore, as in this case, clinical symptoms may show improvement, so brace treatment for scoliosis may be considered.
This patient's symptoms improved somewhat with asthma medications, but they showed a wax-and-wane pattern. However, after starting brace treatment, the Cobb’s angle improved in outpatient follow-up observations, and most importantly, the number of visits to medical institutions due to acute dyspnea symptoms decreased sharply. This is thought to be because the scoliosis angle was alleviated and the force that was compressing the right bronchus intermedius was relieved due to brace treatment.
Most cases of scoliosis show aspects of obstructive pulmonary disease caused by structural pressure, but cases showing acute worsening of asthma like this case are not common. Based on various test results, this patient is thought to be a case with a combination of both obstructive and restrictive pulmonary disease, and it was confirmed that the symptoms actually improved with both brace and drug treatment.
In the case of the patient, the methacholine challenge test was not performed because baseline FEV1 value was less than 60% of the predicted value, as it was contraindicated according to the Ministry of Food and Drug Safety guidelines [12].
Notes
Conflicts of interest
No potential conflict of interest relevant to this article was reported.
Funding
None.
Author contributions
Conceptualization: JKK, YHH. Data curation: JKK; Investigation: JKK. Methodology: JKK, YHH. Project administration: JKK. Resources: JKK, YHH. Supervision: YHH. Visualization: JKK. Writing – original draft: JKK. Writing – review & editing: JKK, YHH.
