Multiple pulmonary leiomyomatous hamartoma with secondary ossification

Kato, Noriko; Endo, Yasushi; Tamura, Gen; Motoyama, Teiichi

doi:10.1046/j.1440-1827.1999.00850.x

Cited by 14 publications

(19 citation statements)

References 8 publications

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“…Indeed, comparative genomic hybridization and X‐chromosome inactivation analysis has revealed the monoclonal origin of both uterine and pulmonary tumors, indicating that the pulmonary lesions actually represent metastasis from the uterine leiomyoma 5 . But some cases, both in which hormones were or were not responsible, without pre‐existing smooth‐muscle tumor, have also been reported 6–8 . Of these, at least two were apparently considered as true fibroleiomyomatous hamartoma, originating from the lung, 4,8 although the authors diagnosed the lesions as leiomyoma 4 .…”

Section: Discussionmentioning

confidence: 99%

“…Indeed, in a recent report, comparative genomic hybridization and X‐chromosome inactivation analysis of a case of benign metastasizing leiomyoma revealed the monoclonal origin of both uterine and pulmonary tumors, indicating that the pulmonary lesions actually represented metastasis from the uterine leiomyoma 5 . However, some cases, both those in which hormones were or were not responsible, without pre‐existing smooth‐muscle tumor, have also been reported 6–8 . These results suggest that the diagnostic term of fibroleiomyomatous hamartoma or benign metastasizing leiomyoma includes two different conditions; one in which the tumor has actually metastasized from a pre‐existing smooth‐muscle tumor and another which is a ‘true’ hamartoma or benign pulmonary muscular proliferation arising primarily in the lung.…”

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confidence: 98%

“…5 However, some cases, both those in which hormones were or were not responsible, without pre-existing smooth-muscle tumor, have also been reported. [6][7][8] These results suggest that the diagnostic term of fibroleiomyomatous hamartoma or benign metastasizing leiomyoma includes two different conditions; one in which the tumor has actually metastasized from a pre-existing smooth-muscle tumor and another which is a 'true' hamartoma or benign pulmonary muscular proliferation arising primarily in the lung. The two conditions are histologically indistinguishable from each other.…”

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confidence: 98%

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Solitary fibroleiomyomatous hamartoma of the lung in a patient without a pre‐existing smooth‐muscle tumor

Itoh

Yanagi

Setoyama

et al. 2001

Pathology International

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A solitary well-demarcated tumor was found in the left lung of a 53-year-old man. It was located in the posterior region of the lower lobe just adjacent to, but apart from, the pleura. It was resected by video-associated thoracic surgery. Macroscopically, the tumor was a whitish solid nodule without hemorrhage or necrosis, and it was 1.5 cm in diameter. Histologically, the tumor consisted of a proliferation of fibromuscular tissue in interlacing fascicles in which many tubular or cleft-like epithelial inclusions were involved. The epithelial inclusions showed cystic changes with goblet cell metaplasia in part, but no atypical changes. Other mesenchymal components such as cartilaginous, myxomatous or adipose tissues were not seen. The patient had no history of neoplasm, including smooth-muscle tumor. Thus, we diagnosed this tumor as a "true" fibroleiomyomatous hamartoma, as distinct from so-called fibroleiomyomatous hamartoma or benign metastasizing leiomyoma, which are usually found in the lungs of women who have had hysterectomies, as multiple fibromuscular nodules. We report here this rare case and we review and discuss published reports of fibromuscular tumors of the lung.

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Section: Discussionmentioning

confidence: 99%

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confidence: 98%

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confidence: 98%

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Solitary fibroleiomyomatous hamartoma of the lung in a patient without a pre‐existing smooth‐muscle tumor

Itoh

Yanagi

Setoyama

et al. 2001

Pathology International

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“…The localized type refers to focal deposition of bone within or adjacent to lung tissue injured by any kind of disease process, mainly including lung abscess, tuberculosis and other infectious, or parasitic diseases, trauma, primary tumors, and metastatic cancer. [7][8][9][10][11][12][13][14][15][16][17] The diffuse type refers to the disseminated formation of spicules or nodules of bone in the interalveolar, interlobular, and subpleural connective tissue of the entire lung and in the alveolar spaces. [2][3][4][5]18,19 Two types of diffuse pulmonary ossification are recognized: the nodular, circumscribed, or tuberous type, and the dendriform, branching, racemose, or trabecular type.…”

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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“…In 1999 we reported on a 31‐year‐old woman with multiple pulmonary leiomyomatous hamartoma (MPLH) 1 . MPLH is a peculiar lesion of the lung, the individual lesions of which are composed of a mixture of smooth muscle and glandular elements 2 .…”

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confidence: 99%