Dendriform Pulmonary Ossification

Anan, Keisuke; Hisanaga, Jumpei; Kawamura, Kodai

doi:10.2169/internalmedicine.1764-18

Cited by 4 publications

(4 citation statements)

References 2 publications

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“…Other secondary cases of DPO were observed in patients in their 60s–80s 32 33. However, recent reports have shown that IDPO diagnosed by surgical lung biopsy was distributed among patients in their 20s–40s 16–18 25–29. In our study, the diagnosis was made in 5 patients in their 20s and 8 patients in their 30s.…”

Section: Discussioncontrasting

confidence: 59%

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Nationwide retrospective observational study of idiopathic dendriform pulmonary ossification: clinical features with a progressive phenotype

et al. 2022

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BackgroundDiffuse pulmonary ossification is a specific lung condition that is accompanied by underlying diseases. However, idiopathic dendriform pulmonary ossification (IDPO) is extremely rare, and the clinical features remain unclear. In this study, we aimed to report the clinical characteristics of IDPO.MethodsWe conducted a nationwide survey of patients with IDPO from 2017 to 2019 in Japan and evaluated the clinical, radiological, and histopathological findings of patients diagnosed with IDPO.ResultsTwenty-two cases of IDPO were identified. Most subjects (82%) were male, aged 22–56 years (mean (SD), 37.9 (9.1)) at diagnosis. Nearly 80% of the subjects were asymptomatic, and the condition was discovered during a medical check-up. However, 36% of the subjects showed a decline in forced vital capacity (%FVC) predicted <80% at diagnosis. The typical radiological features of high-resolution CT (HRCT) are calcified branching structures that are predominantly distributed in the lower lung fields without any other conspicuous finding. Histopathological analysis also showed dendriform ossified lesions from the intraluminal areas to interstitial areas. Notably, during the follow-up period of 20 years, disease progression was found in 88% on HRCT and more than 50% on pulmonary function tests (FVC and/or forced expiratory volume in 1 s). Two cases with rapid decline of 10% /year in %FVC predicted were observed.)) at diagnosis. Nearly 80% of the subjects were asymptomatic, and the condition was discovered during a medical check-up. However, 36% of the subjects showed a decline in forced vital capacity (%FVC) predicted <80% at diagnosis. The typical radiological features of high-resolution CT (HRCT) are calcified branching structures that are predominantly distributed in the lower lung fields without any other conspicuous finding. Histopathological analysis also showed dendriform ossified lesions from the intraluminal areas to interstitial areas. Notably, during the follow-up period of 20 years, disease progression was found in 88% on HRCT and more than 50% on pulmonary function tests (FVC and/or forced expiratory volume in 1 s). Two cases with rapid decline of 10% /year in %FVC predicted were observed.ConclusionsIDPO develops at a young age with gradually progressive phenotype. Further research and long-term (>20 years) follow-up are required to clarify the pathogenesis and clinical findings in IDPO.

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Section: Discussioncontrasting

confidence: 59%

“…All patients were diagnosed by surgical lung biopsy. Among the 22 patients, the cases of nine patients have already been reported in various journals 12 24–29…”

Section: Resultsmentioning

confidence: 99%

Nationwide retrospective observational study of idiopathic dendriform pulmonary ossification: clinical features with a progressive phenotype

et al. 2022

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“…4,5 Four cases were diagnosed by SLB because these patients could not be definitively diagnosed by TBLB. 2,[6][7][8] Thus, the diagnostic accuracy of TBLB for DPO, as well as in other interstitial lung diseases, may be controversial.…”

Section: Discussionmentioning

confidence: 99%

Idiopathic dendriform pulmonary ossification diagnosed by bronchoscopic lung cryobiopsy: A case report

Sekimoto

Nagata

Kato

et al. 2021

Respirology Case Reports

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Dendriform pulmonary ossification (DPO) is a rare condition characterized by heterotopic bone production of unknown origin within the pulmonary tissue. Many cases are asymptomatic with slow progression and are often diagnosed incidentally during autopsy. Thus, only few cases are diagnosed while the patient is still alive since surgical lung biopsy is often needed for pathological diagnosis. This is the case of a 37-yearold man treated at our hospital due to abnormal findings on chest x-ray without any symptoms. His high-resolution computed tomography revealed diffuse reticular shadows and micronodules, consistent with calcification. He underwent transbronchial lung cryobiopsy (TBLC) and was diagnosed with idiopathic DPO based on pathological findings. To our knowledge, this is the first reported case of DPO diagnosed using TBLC. TBLC can be a useful yet minimally invasive diagnostic tool to diagnose DPO and other interstitial lung diseases.

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“…Tseung et al reported that the frequency of DPO was 0.16% and that the median patient age was 72 (range 32-91) years old, with 88% of affected individuals being men (2). Recently, cases of DPO in younger people have been identified during unrelated medical examinations and thus are diagnosed while the individuals are still alive (6,7).…”

Section: Discussionmentioning

confidence: 99%

Diffuse Pulmonary Ossification with Connective Tissue Weakness Potentially Due to Vascular Ehlers-Danlos Syndrome

et al. 2021

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A 30-year-old non-smoking man was referred to our hospital for the further examination of abnormal shadows revealed by chest X-ray. He had mild shortness of breath. Chest computed tomography revealed a fine-grained dendritic shadow with diffuse calcification in both lungs and as well as emphysematous changes in the upper lung lobes. A surgical lung biopsy histology revealed diffuse pulmonary ossification complicated with lung laceration, vascular disruption, hemosiderosis, and emphysema, suggesting vascular Ehlers-Danlos syndrome (vEDS). However, the patient had no external physical signs or family history of vEDS and no COL3A1 gene mutations. We are closely monitoring this patient in the clinic.

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Dendriform Pulmonary Ossification

Cited by 4 publications

References 2 publications

Nationwide retrospective observational study of idiopathic dendriform pulmonary ossification: clinical features with a progressive phenotype

Nationwide retrospective observational study of idiopathic dendriform pulmonary ossification: clinical features with a progressive phenotype

Idiopathic dendriform pulmonary ossification diagnosed by bronchoscopic lung cryobiopsy: A case report

Diffuse Pulmonary Ossification with Connective Tissue Weakness Potentially Due to Vascular Ehlers-Danlos Syndrome

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