Asymmetrical Interstitial Lung Disease Suggested to Be Due to Hypoplasia of the Unilateral Pulmonary Artery: A Case Report with a 20-year Follow-up

Tomioka, Haruaki; Amimoto, Hisanori; Fujii, Hiroshi; Katsuyama, Eiji; Okuno, T; Kawabata, Yoshinori

doi:10.2169/internalmedicine.5753-20

Cited by 2 publications

(2 citation statements)

References 13 publications

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“…At present, only some UAPA cases diagnosed by echocardiography have been reported [9,10]. These reports comfirmed the diagnostic value of echoardiography in UAPA, and analyzed the complications caused by UAPA [11,12], and used for follow up assessment after surgery [13]. However, the clinical understanding of this disease is limited, and few references are focusing on the echocardiographic characteristics, changes of pulmonary artery pressure and prognosis.…”

Section: Introductionmentioning

confidence: 99%

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Unilateral absence of pulmonary artery analysis based on echocardiographic feature

Tian¹,

Zheng²

2021

Reviews in Cardiovascular Medicine

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The unilateral absence of pulmonary artery (UAPA) is a rare congenital cardiovascular malformation, which is asymptomatic and easy to be ignored in early stage. A large number of complications may occur in the later stage. Therefore, early diagnosis and treatment is of great significance. The imaging data of 49 patients with UAPA discovered and confirmed clinically by the echocardiography in our hospital are analyzed. The results show that left pulmonary artery absence is more common (55%) and most of them are associated with other cardiovascular malformations (92%). Atrial septal defect and patent foramen ovale were most common in 56%. In which the absence of isolated pulmonary artery was 8% (4/49), and the absence of right pulmonary artery was 75% (3/4). Especially in the patients with tetralogy of Fallot, 77% (5/9) of them miss the diagnosis of UAPA. This suggests that doctors and sonographers should pay attention to the development of pulmonary artery bifurcation and left and right branches in multi-section, and strengthen the scanning of short axis section of high large artery.

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Section: Introductionmentioning

confidence: 99%

“…The early clinical manifestations of UAPA are lack of specificity [2]. The symptoms are not typical, often with repeated pulmonary infection, hemoptysis, dyspnea, and easy to miss diagnosis and misdiagnosis [3].…”

Section: Introductionmentioning

confidence: 99%

Unilateral absence of pulmonary artery analysis based on echocardiographic feature

Tian¹,

Zheng²

2021

Reviews in Cardiovascular Medicine

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Chest Computed Tomography Findings in Unilateral Pulmonary Fibrosis Secondary to Chronic Hypoperfusion

Marrocchio,

Humphries,

Lynch

2023

Journal of Thoracic Imaging

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Purpose: Unilateral lung fibrosis is uncommon and few cases secondary to parenchymal hypoperfusion have been reported, requiring further understanding of this entity. This study aims to report the chest computed tomography (CT) findings of patients with unilateral lung fibrosis related to parenchymal hypoperfusion observed in our institution. Patients and Methods: Patients with a chest CT between 2004 and 2022 showing a condition causing hypoperfusion of either lung and ipsilateral unilateral lung fibrosis were retrospectively identified. Clinical and scintigraphic data were collected. Pattern and distribution of fibrosis were recorded, and its progression was evaluated when follow-up was available. In adequate CTs, fibrosis was quantified using data-driven textural analysis (DTA). Affected and contralateral lungs and baseline and follow-up data were compared using the Wilcoxon signed-rank test. Results: Thirteen patients (male: 7, female: 6, median age: 61 y) were included; 5 with congenital unilateral absence of a pulmonary artery and 8 with fibrosing mediastinitis. The mean scintigraphic perfusion of affected lungs was 3.3% ± 1.1 compared with 96.7% ± 1.1 contralaterally (n = 7, P = 0.017). Fibrosis had a UIP pattern in one case, indeterminate in the others, and was most commonly diffuse craniocaudally and peripheral or central axially. DTA in 12 patients showed a mean fibrotic score of 32% ± 24.6 compared with 0.5% ± 0.4 in the contralateral lungs (P = 0.002). Median follow-up was 4.5 years (minimum to maximum: 1 to 13 y). Of 10 patients, fibrosis was progressive in 60%. DTA of 5 follow-up CTs showed increased reticulations (P = 0.043). Conclusion: In patients with lung hypoperfusion, the possible complication of lung fibrosis should be considered.

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Asymmetrical Interstitial Lung Disease Suggested to Be Due to Hypoplasia of the Unilateral Pulmonary Artery: A Case Report with a 20-year Follow-up

Cited by 2 publications

References 13 publications

Unilateral absence of pulmonary artery analysis based on echocardiographic feature

Unilateral absence of pulmonary artery analysis based on echocardiographic feature

Chest Computed Tomography Findings in Unilateral Pulmonary Fibrosis Secondary to Chronic Hypoperfusion

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