Pulmonary sclerosing pneumocytoma remains a diagnostic challenge using frozen sections: a clinicopathological analysis of 59 cases

Yang, Cheng‐Han; Lee, Li‐Yu

doi:10.1111/his.13391

Cited by 32 publications

(29 citation statements)

References 18 publications

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“…However, some atypical cases showed either different clinical behaviors from typical PSP, such as recurrence or metastasis, or confused histological characteristics, such as limited typical patterns, cytological atypia, and focal necrosis. These atypical cases could be misdiagnosed as papillary or solid subtype of lung adenocarcinoma and neuroendocrine tumor, especially in the case of needle biopsy or intraoperative frozen diagnosis [21]. In this case, the delayed diagnosis at biopsy was mainly attributed to the atypical phenomenon, including multiple LN metastases, papillary and solid growth pattern, cellular atypia, tumoral necroticfoci, and higher Ki-67 LI than that of ordinary PSP cases.…”

Section: Discussionmentioning

confidence: 85%

Clinicopathological and Molecular Characteristics of Atypical Pulmonary Sclerosing Pneumocytoma With Multiple Metastases

Wang¹,

Yang²,

Jiang³

et al. 2020

Preprint

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Background: Few patients with pulmonary sclerosing pneumocytoma (PSP) may suffer from recurrence and oligometastasis as it is a benign tumor or has a low malignancy potential. Herein, an in-depth study, based on an extremely rare PSP case with atypical features, was carried out to elaborate the potential mechanism underlying the rapid malignant progression. Methods: The clinicopathological data of this atypical PSP (AP) case were obtained. Formalin‑fixed and paraffin‑embedded tissues from all the lesions of AP and five classic PSP cases (as control) were used for whole-exome sequencing (WES) and immunohistochemistry. Results: A 23-year-old male showed a 6.5-cm pulmonary nodule in the right middle lobe (RML) and enlarged mediastinal lymph nodes (LNs). He underwent thoracoscopic RML lobectomy, systematic LNs dissection, and mediastinal lymphadenectomy. The metastases to the cervical LNs and liver were detected in a short period and then resected. Postoperative pathological examination confirmed the diagnosis of PSP in all the lesions, based on the typical histological characteristics and immunophenotypes. Furthermore, WES identified both AKT1 E17K somatic mutation and TP53 C176Y germline mutation in this AP case. The genomic evolution analysis showed different evolutionary branches in the metastatic tumors distinct from the primary lesion. Moreover, compared to the control group, the AP case showed high copy number variations (CNVs) and significantly high copy number instability (CNI). Conclusions: We speculated that the atypical histopathological features and malignant behaviors may be due to the co-mutations of somatic AKT1 E17K and germline TP53 C176Y, combined with the high CNVs and CNI.

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Section: Discussionmentioning

confidence: 85%

Clinicopathological and Molecular Characteristics of Atypical Pulmonary Sclerosing Pneumocytoma With Multiple Metastases

Wang¹,

Yang²,

Jiang³

et al. 2020

Preprint

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“…Intraoperative frozen section analysis is prevalently used to assess resection margins during cancer surgery, and is a determinant whether addition resection is warranted to achieve R0 [50,51]. But the clinical value of intraoperative frozen section in pCCA was limited, because the sensitivity was reported to be only 68% [21].…”

Section: Plos Onementioning

confidence: 99%

Does additional resection of a positive microscopic ductal margin benefit patients with perihilar cholangiocarcinoma: A systematic review and meta-analysis

Chen

Huang

et al. 2020

PLoS ONE

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Background The incidence of a positive microscopic ductal margin (R1) after surgical resection for perihilar cholangiocarcinoma (pCCA) remains high, but the beneficial of additional resection has not been confirmed by any meta-analysis and randomized clinical trials (RCT), which also increased the risk of morbidity and mortality. Hence, a systematic review is warranted to evaluate the clinical value of additional resection of intraoperative R1 for pCCA. Methods Eligible studies were searched by PubMed, MedLine, Embase, the Cochrane Library, Web of Science, from Jan.1 st 2000 to Nov.30 th 2019, evaluating the 1-, 3-, and 5-year overall survival (OS) rates of additional resection of intraoperative pathologic R1 for pCCA. Odds ratio (OR) with 95% confidence interval (CI) was used to determine the effect size by a randomized-effect model. Results Eight studies were enrolled in this meta-analysis, including 179 patients in the secondary R0 group, 843 patients in the primary R0 group and 253 patients in the R1 group. The pooled OR for the 1-, 3-, and 5-year OS rate between secondary R0 group and primary R0 group were 1.03(95%CI 0.64~1.67, P = 0.90), 0.92(95%CI 0.52~1.64, P = 0.78), and 0.83(95%CI 0.37~1.84, P = 0.65), respectively. The pooled OR for the 1-, 3-, and 5-year OS rate between secondary R0 group and R1 group were 2.14(95%CI 1.31~3.50, P = 0.002), 2.58 (95%CI 1.28~5.21, P = 0.008), and 3.54(95%CI 1.67~7.50, P = 0.001), respectively. However, subgroup analysis of the West showed that the pooled OR for the 1-, and 3-year OS

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“…It was later found to be a tumor in the lung parenchyma that originated from type II alveolar epithelial cells and had a successful clinical process (2,3). World Health Organization (WHO) (2015) has histologically classified lung tumors and renamed it as pulmonary sclerosing pneumocytoma and identified as a lung adenoma (4). PSP has been reported as the most common benign tumor in the lung, while the incidence rate was relatively high in East Asia.…”

Section: Introductionmentioning

confidence: 99%

“…More than 80% of cases occurred in middle-aged women > 50 years of age (5,6). Since most PSPs were discovered by accident, there were no typical reliable clinical manifestations, and the accuracy rate of the intraoperative frozen section (FSS) was 44.1%, while the delay rate was 15.3% (4). Despite of benign nature, PSP represents a diagnostic challenge due to its controversial etiology and biologic behavior, as well as the diversity of pathohistological findings.…”

Section: Introductionmentioning

confidence: 99%

Correlation Between Pulmonary Sclerosing Pneumocytoma Features and MSCT Imaging Manifestations in 34 Patients: Implications for Precision Medicine

Wang

Xiong

et al. 2021

Front. Med.

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Objective: To identify and analyze the multi-slice computed tomography (MSCT) imaging manifestations and clinicopathological features of PSP to improve the preoperative and intraoperative diagnosis of the disease.Method: This was a retrospective study conducted on the imaging and clinicopathological data of the PSP patients treated in two major hospitals in China from October 2001 to December 2019. The locations of lung lesions, clinical symptoms, surgical complications, MSCT imaging features, and the corresponding relationship with clinicopathological features were assessed. Then, a new diagnostic approach was defined and used to train imaging and pathological doctors (experimental group). Then, the diagnostic accuracy of the experimental group was evaluated in preoperative and intraoperative diagnosis of PSP.Results: Thirty-four PSP cases were analyzed (mean: 51.42; range: 39–69 years old). The peripheral type was more common, while 92% of the lesions located in the middle lobe of the right lung and the lower lobe of bilateral lungs. The shortest lesion edge-pleura distance ranged 0 to 30 mm and 46% of the lesions (16/34) were attached to the pleura, 62% (21/34) located at 0–5 mm, 92% (31/34) within 20 mm from the pleura. Diameters of the lesions ranged 8.58 to 68.41 mm, while most of them were 20-40 mm. All lesions showed enhancement, and 97% (33/34) were unevenly enhanced. PSP volume was negatively correlated with the total degree of enhancement (r = −0.587, p < 0.01), and the volume difference between the obvious enhancement zone and the slight enhancement zone (r = −0.795, p < 0.01). Welt vessel sign was observed in 61.7% (21/34) of cases, and none of welt vessels entered into the lesions. Vascular-like enhancement area inside the lesion showed no significant correlation with the welt vessels outside the lesion, and no case showed entrance of bronchus into the lesion. The trained experimental group showed significantly greater diagnostic accuracy than the control group. In particular, the accuracy rate of intraoperative frozen section diagnosis was 60% higher in the experimental group than the control group.Conclusion: PSP has characteristic imaging manifestations, which can be utilized to improve the preoperative and intraoperative diagnostic coincidence rate of PSP.

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Pulmonary sclerosing pneumocytoma remains a diagnostic challenge using frozen sections: a clinicopathological analysis of 59 cases

Abstract: In addition to evaluating the tumour circumscription and identifying the various growth patterns, we propose that the key to avoiding a misdiagnosis is to recognise the dual-cell populations in a tumour, i.e. cuboidal surface cells and stromal round cells.

Cited by 32 publications

References 18 publications

Clinicopathological and Molecular Characteristics of Atypical Pulmonary Sclerosing Pneumocytoma With Multiple Metastases

Clinicopathological and Molecular Characteristics of Atypical Pulmonary Sclerosing Pneumocytoma With Multiple Metastases

Does additional resection of a positive microscopic ductal margin benefit patients with perihilar cholangiocarcinoma: A systematic review and meta-analysis

Correlation Between Pulmonary Sclerosing Pneumocytoma Features and MSCT Imaging Manifestations in 34 Patients: Implications for Precision Medicine

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