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 may be involved, such as genetic effects, congenital structural abnormalities, neuromuscular diseases, hormonal imbalance, and asymmetric growth. Severe scoliosis requiring surgical treatment defined as a Cobb’s 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 exempted from review by the Institutional Review Board (IRB) of Busan St. Mary’s Hospital (IRB No: 24-14000-134). Written informed consent was obtained from both the patient and the legal guardian after a thorough explanation of the study.

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 adolescent 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. At the time of dyspnea, tachypnea was accompanied, but oxygen administration was not required, and there were no restrictions on daily life, but shortness of breath occurred during exercise. For the results of the pulmonary function tests, refer to Table 1 (conducted on April 8, 2023).

But a chest X-ray (CXR) shows no increased opacities or consolidation/hyperinflation. Based on the clinical symptoms, the researchers empirically diagnosed bronchial asthma in December 2022 and treated the patient with short-acing beta agonist, short-acting muscarinic antagonist, leukotriene receptor antagonist, oral steroids and inhaled corticosteroid agents (Table 2) for about 6 months, however she did not show significant improvement in her symptoms. The patient visited the emergency room 3 times at night, was hospitalized 3 times, and received outpatient treatment several times for acute dyspnea over a period of approximately 6 months since December 2022. Eosinophil, serum immunoglobulin E, hemoglobin, O₂ saturation, ImmunoCAP (inhalant), gas analysis values (Tables 3, 4), and transthoracic echocardiogram results were normal at presentation.

While continuing outpatient treatment, we recognized that the Cobb’s angle on CXR corresponds to 70°. 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, 1D). Despite abouth 6 months of drug treatment, the symptoms did not improve much, 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 chest CT scan was not performed because the patient refused it.

The patient originally had a prescribed brace, but rarely used it due to the discomfort of wearing it for a long time. However, after the cause of bronchial compression was identified through a chest CT scan, the patient’s compliance with treatment increased, and the wearing time gradually increased with a new and more comfortable scoliosis brace. In the meantime, the number of visits to the emergency room visits and hospitalizations decreased significantly, and the 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% (Fig. 1C, 1D, Table 1), and she has no significant limitations in her daily life.

Discussion

In this case, the physicians 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 height 155 cm, weight 40 kg, body mass index 16.65 (5th percentile, underweight), who had never been hospitalized for pneumonia except during childhood, and had no history of respiratory problems such as interstitial lung disease. There is no evidence of nutritional imbalance and secondary sexual characteristics have developed normally. Additionally, there is no past medical history or family history suggestive of ankylosing spondylitis, multiple sclerosis, myasthenia gravis, etc.

Over the past few years, as the COVID-19 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 medications, but the pattern of relapse when medications were discontinued was repeated. However, the number of outpatient and emergency room visits due to acute dyspnea decreased rapidly with the use of new brace from June 2023. The patient’s last emergency room visit was on April 5, 2023, and the symptoms could be controlled through outpatient follow-up observations. The Cobb’s angle on CXR also began to improve (Fig. 1C, 1D). This is presumed to be because the Cobb’s angle decreased by the scoliosis brace, thereby relieving the pressure on the right bronchus intermedius.

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 (2020).

Article information

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. All authors read and approved the final manuscript.

Fig. 1.

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°).

Fig. 2.

Compression of the right bronchus intermedius by 8th thoracic vertebra confirmed on contrast-enhanced chest computed tomography. (A) Horizontal plane. (B) Coronal plane.

References

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Figure & Data

References

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