A Rare Case of Asymptomatic Diffuse Pulmonary Ossification Detected during a Routine Health Examination

Mizushina, Yoshiko; Bando, Masashi; Hosono, Tatsuya; Mato, Naoko; Nakaya, Takakiyo; Yamasawa, Hideaki; Hironaka, Mitsugu; Tanaka, Akira; Sugiyama, Yukihiko

doi:10.2169/internalmedicine.51.7520

Cited by 16 publications

(11 citation statements)

References 7 publications

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“…In the present study, we also found more ossifications in the posterior portion of the right lower lobe with respect to the anterior one. Posterior predilection of DPO has been reported by Mizushina et al . and is probably explained in the present case by the fact that the patient was bedridden.…”

Section: Discussionsupporting

confidence: 66%

Diffuse pulmonary ossification in permanent vegetative state

Porzionato

Macchi

Broi

et al. 2014

Pathology International

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Diffuse pulmonary ossification (DPO) is a rare condition characterized by diffuse metaplastic bone formation in the lungs. Two patterns have been described: dendriform, with a coral-like network of bone spiculae along the alveolar septa; and nodular, with lobulated fragments in the alveolar spaces. Dendriform DPO is frequently associated with chronic pulmonary pathologies. We present here the first case of DPO associated with a long-lasting vegetative state. Micro-computed tomography (MicroCT) was applied to analyze the distribution of pulmonary ossification in volumes of lung samples. It showed a mean volume percentage of ossification of 0.79% and 3-D reconstructions permitted to reveal the branching pattern and internal cavities of some ossifications. The occurrence of DPO in a persistent vegetative state (PVS) may be favored by recurrent pulmonary infections, due to aspiration and immunological defects, and respiratory instability, due to brain damage and the fact of being bedridden. Fibrotic reactions probably represent a preliminary step in bone formation. Further studies could examine the incidence and clinical significance of DPO in subjects in PVS or patients who are bedridden for other reasons. MicroCT may facilitate analysis of more case histories, with greater sensitivity with respect to classic microscopic analyses.

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Section: Discussionsupporting

confidence: 66%

Diffuse pulmonary ossification in permanent vegetative state

Porzionato

Macchi

Broi

et al. 2014

Pathology International

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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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“…92 A few patients were diabetic 6, 40(case 6), 41(4 cases),50 and 1 case was acromegalic. 86 Less than 10 cases were identified to be truly idiopathic in the absence of any underlying pulmonary disease, 46,49,53,63,64,81,88,91 and 2 cases occurred in the same family. 46 Excluded from this counting are about 20 clinicopathologic overlap cases (as previously defined) between the nodular and dendriform forms of diffuse pulmonary ossification 32(7 cases), 39(6 cases), 56(case 7) , as well as 7 other overlap cases of diffuse nodular pulmonary ossification published from 3 different sources in association with Hamman-Rich syndrome, all cited by Mendeloff,79 and the case reported by Popelka and Kleinerman, 100 which occurred in a lung shock after heart surgery and prolonged mechanical ventilation.…”

Section: Authors' Commentmentioning

confidence: 99%

Selected Case From the Arkadi M. Rywlin International Pathology Slide Series

Bisceglia¹,

Chiaramonte

Panniello

et al. 2015

Advances in Anatomic Pathology

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Two cases of diffuse dendriform pulmonary ossification (DPO) are presented, one of the secondary type and the other of the idiopathic type. Case 1 was an adult female patient who underwent thoracic surgery to remove a posterior mediastinal bronchogenic cyst, which was discovered on a computed tomography scan performed after an episode of pneumonia when traction bronchiectasis with interstitial lung disease/fibrosis was also suspected in the lungs. Histologic examination performed on the resected lung tissue revealed numerous scattered small osseous spicules on a background of intense interstitial inflammation and fibrosis, leading to further clinical and laboratory investigations and the final diagnosis of DPO secondary to lung involvement by scleroderma. Case 2 was an adult male patient who underwent thoracoscopic exploration after a computed tomography scan, which revealed traction bronchiectasis with linear thickening of the interstitial lung tissue. Histologic examination of the lung tissue specimen revealed numerous osseous spicules in the absence of interstitial septal inflammation. Noteworthy in this case were also some nodules of collagenized tissue similar to those seen in the lungs of patients affected by Ehlers-Danlos syndrome. The absence of any clinical sign or symptoms related to Ehlers-Danlos syndrome attest to the nonspecificity of these pulmonary fibrous nodules. No case of DPO secondary to scleroderma has been reported in the literature so far, although around half of the patients with scleroderma manifest pulmonary diseases. Idiopathic DPO is even rarer, usually discovered postmortem, with only 20 cases diagnosed in life with lung biopsies taken by open surgery or through a thoracoscopic approach. DPO is often misdiagnosed radiologically as bronchiectasis and/or interstitial lung disease/fibrosis.

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A Rare Case of Asymptomatic Diffuse Pulmonary Ossification Detected during a Routine Health Examination

Cited by 16 publications

References 7 publications

Diffuse pulmonary ossification in permanent vegetative state

Diffuse pulmonary ossification in permanent vegetative state

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

Selected Case From the Arkadi M. Rywlin International Pathology Slide Series

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