A rare pulmonary hamartoma: fibroleiomyomatous hamartoma

Ichiki, Yoshinobu; Kawasaki, Junji; Hamatsu, Takayuki; Suehiro, Taketoshi; Shibuya, Ryo; Matsuyama, Atsuji; Tanaka, Fumihiro; Hisaoka, Masanori; Sugimachi, Keizō

doi:10.1186/s40792-016-0184-z

Cited by 9 publications

(13 citation statements)

References 4 publications

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“…the PFLH is only composed of ber and smooth muscle tissue and invaginated respiratory epithelium. The lesion is often adjacent to the bronchioles or thick wall vessels, or has thick wall vessels or bronchial epithelium penetrating into the leision [3]. In our case, the bronchial epithelium extended into the alveolar septa in the pulmonary parenchyma without a cartilaginous or adipose element.…”

Section: Discussionmentioning

confidence: 63%

Three unusual Pulmonary fibroleiomyomatous hamartomas: cases report and literature review

Song

Wang

Fu³

2021

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BackgroundPulmonary fibroleiomyomatous hamartomas (PFLH) is an extremely rare benign leision, characterized by most patients have no respiratory symptoms and pulmonary nodules is found during rutine chest imaging. It’s completely different from fibrochondrolipoma (ie.chondrogenic pulmonary hamartoma, FCL), Herein, we report three unusual cases of PFLH in a 48-year-old male patient, a 63-year-old male patient and a 63-year-old female patient,respectively and discuss their morphological characteristics and differential diagnoses including fibrochondrolipoma (FCL), pulmonary fibroadenoma (PFA), primary pulmonary leiomyoma (PPL), bronchioloalveolar adenoma (BAA), solitary fibrous tumor (SFT) and pulmonary metastatic leiomyoma (PML) in female patients.Case presentationThree cases showed singler and multiple nodules in the pulmonary parenchyma,the boundary of these nodules was clear. it showed swelling growth and lobulated in larger tumors. it consisted of the mesenchymal rich in spindle cells and the glandular epithelium tissue. the spindle cells form smooth muscle tissue and fibrous tissue, the glandular epithelium with invagination showed glandular ducts or fissures, disorder of composition. Immunohistochemical staining showed Desmin, SMA,H-caldesmon-positive in the all spindle cells; H-caldesmon-positive, SMA-weak positive and desmin-negative in the long spindle cells; TTF-1,CK7 were positive in glandular epithelium and P63,CK5/6 were expressed in basal cells; HMB45,S-100,CD34, ER,PR were negative in all cells. Additionally, Ki-67 VPI was low and less than 1%. The immunohistochemical analysis combined with the morphological results supported the diagnosis of solitary PFLH (SPFLH) or multiple PFLH (M PFLH). There was no recurrence in SPFLH and recurrence in MFLH in the group.ConclusionsPFLH is a rare benign leision .It originated from the small bronchus or smooth muscle cells of vascular wall which implanted into the lung parenchyma and overgrowth into nodules or mass, the messenchymal component of PFLH is the most important for accurate pathological diagnosis, PFLH is easy to be confused with PFA and PPL when there are not enough immune marker such as SMA,desmin, H-caldesmon to mark the mesenchymal components. Therefore, Diagnosis of PFLH mainly depends on pathological examination, females patients have to exclude PML.

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Section: Discussionmentioning

confidence: 63%

Three unusual Pulmonary fibroleiomyomatous hamartomas: cases report and literature review

Song

Wang

Fu³

2021

Preprint

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“…So far, it is only ve patients with solitary pulmonary broleiomyomatous hamartoma had been reported [2]. The histological diagnosis criteria are "a wellcircumscribed nodule composed of smooth muscle cells with numerous epithelial inclusions" and "no cellular atypia or mitotic gures" [2]. While in this present clinical case we showed a huge lung broleiomyomatous hamartoma found in pleural cavity, which was rst misdiagnosed as germ cell tumor with pleural effusion considered as pleural metastasis.…”

Section: Introductionmentioning

confidence: 48%

“…It is an extremely rare type of benign tumor could be found in every sites of lung. So far, it is only ve patients with solitary pulmonary broleiomyomatous hamartoma had been reported [2]. The histological diagnosis criteria are "a wellcircumscribed nodule composed of smooth muscle cells with numerous epithelial inclusions" and "no cellular atypia or mitotic gures" [2].…”

Section: Introductionmentioning

confidence: 99%

A Misdiagnosis of a Huge Lung Fibroleiomyomatous Hamartoma in Pleural Cavity

Liu

Zhang

et al. 2021

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Backgroud: Lung fibroleiomyomatous hamartoma is an extremely rare benign tumor in lung, it belongs to hamartoma. There were only five patients with solitary pulmonary fibroleiomyomatous hamartoma reported in literatures and our case has the largest size of the tumor from lung.Case presentation: Our case is a 36-year woman with a huge mass in pleural cavity. She was once misdiagnosed mediastinal germ cell tumor with pleural effusion through percutaneous lung biopsy in other hospital. After traditional thoracotomy, a huge size of 22 x 18 x 5.5 cm mass was completely dissected and a diagnosis of pulmonary fibroleiomyomatous hamartoma was given by pathology combined with immunohistochemical staining, which showed the positive staining of SMA, Desmin, CD34, BCL2, TTF1, Napsin A, and CK7, and the negative staining of S-100 and Calretinin. The patient recovered well with 40 months following up without recurrence.Conclusions:In our case, we had reported the largest size of lung fibroleiomyomatous hamartoma. The treatment of fibroleiomyomatous hamartoma is completely resected it and have a good prognosis.

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“…We considered the term bronchial hamartoma or pulmonary hamartoma but opted against these given that hamartoma suggests an abnormal proliferation of normal tissues native to the site of occurrence. In addition, the term pulmonary hamartoma is used in the lung for a benign lesion (also referred to as chondroid hamartoma) that contains various combinations of epithelial and mesenchymal tissues, typically including cartilage and adipose tissue, although a rare variant, known as fibroleiomyomatous 183 BROCHUS-LIKE STRUCTURES IN TERATOMAS hamartoma, has been described which is composed of respiratory epithelium and smooth muscle elements only (15,16). We also considered the term bronchial adenoma but this does not reflect the presence of a smooth muscle component and moreover this term is used interchangeably with mucous gland adenoma in the lung, a benign neoplasm composed of mucinous glands (17,18).…”

Section: Discussionmentioning

confidence: 99%

Bronchus-like Structures in Ovarian Teratomas: Report of a Series of a Previously Unreported Phenomenon With Potential For Misdiagnosis

Rajendran

Boyd

Shah

et al. 2021

International Journal of Gynecological Pathology

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We report 10 cases of a previously undescribed lesion within ovarian teratomas which we designate bronchus-like structures. The lesions occurred in patients aged 16 to 56 yr (mean: 36) and involved the left ovary (n = 5) or right ovary (n = 5). Nine cases were mature teratomas (dermoid cysts/mature cystic teratomas or mature solid teratomas), 1 with somatic malignant transformation, and 1 was an immature teratoma. The bronchus-like structures ranged in size from 2.5 to 10 mm and were unifocal (7 cases) or multifocal (3 cases). The morphology was relatively constant in all cases and characterised by a well-formed bronchus surrounded by glandular structures, some of which were dilated, separated by stroma containing a variable amount of smooth muscle. In all but 1 case, a proportion of the glands contained abundant foamy cytoplasm. There was little or no nuclear atypia or mitotic activity. At low-power, the glands often had a somewhat "infiltrative" appearance and one case was originally diagnosed as a "pulmonary-type" adenocarcinoma arising in a dermoid cyst. In all cases, there was diffuse staining of the bronchus and glands with TTF1 and Napsin A, confirming the cell lineage. Follow-up in 4 cases (18-130 mo; median: 64 mo) showed no evidence of recurrence; 1 patient died from an unrelated malignancy. In reporting this apparently rare but possibly underrecognized benign lesion arising within ovarian teratomas, we discuss the differential diagnosis and stress that pathologists should be aware of this phenomenon in order to avoid an erroneous diagnosis of malignancy.

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A rare pulmonary hamartoma: fibroleiomyomatous hamartoma

Cited by 9 publications

References 4 publications

Three unusual Pulmonary fibroleiomyomatous hamartomas: cases report and literature review

Three unusual Pulmonary fibroleiomyomatous hamartomas: cases report and literature review

A Misdiagnosis of a Huge Lung Fibroleiomyomatous Hamartoma in Pleural Cavity

Bronchus-like Structures in Ovarian Teratomas: Report of a Series of a Previously Unreported Phenomenon With Potential For Misdiagnosis

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