We present a case of a malignant Ewing-like neoplasm of the parotid gland in a 20-year-old woman with an EWSR1-KLF15 gene fusion that presented with pulmonary metastasis. Despite the fact that the ...tumor was essentially immunohistochemically negative for keratins, p63, and p40, we interpret this neoplasm as an unusual form of a high-grade myoepithelial carcinoma based on its focal plasmacytoid cytology, chondromyxoid matrix, SOX10, S100 protein, and calponin expression, and the knowledge that the EWSR1-KLF15 gene fusion has, to date, only been identified in 2 tumors, both myoepithelial carcinomas of the kidney. We also present a cytogenetic analysis of this unusual tumor. This “Ewing-like myoepithelial carcinoma” initially did not respond to 2 cycles of ifosfamide and etoposide alternated with a cycle of cytoxan, adriamycin, and vincristine, a standard regimen for Ewing sarcoma. Subsequent oral pazopanib therapy did result in a reduction of the patient’s pulmonary and nodal disease.
Purpose
To assess the added value of susceptibility-weighted imaging (SWI) to diffusion-weighted imaging (DWI) in the characterization of parotid gland tumors.
Methods
Seventy-eight patients with ...pathologically confirmed parotid gland tumors, who underwent DWI and SWI for pre-surgery evaluation, were enrolled. Apparent diffusion coefficient (ADC) and degree of intratumoral susceptibility signal intensity (ITSS) were measured and compared between benign and malignant groups, and among pleomorphic adenoma (PA), Warthin tumor (WT) and malignant tumor (MT). Independent sample
t
test, one-way analysis of variance and receiver operating characteristic curve analysis were used for statistical analyses.
Results
Benign parotid gland tumor showed a significantly higher mean ADC value than malignant tumors (0.836 ± 0.350 vs 0.592 ± 0.163,
p
= 0.001). Setting an average ADC value of 0.679 as the cut-off value, optimal differentiating performance could be obtained (AUC, 0.700; sensitivity, 62.69%; specificity, 81.82%) for differentiating malignant from benign tumors. PA showed significantly higher mean ADC and less ITSS than WT (ADC,
p
< 0.001; ITSS,
p
= 0.033) and MT (ADC,
p
< 0.001; ITSS,
p
= 0.024), while the difference between WT and MT was not significant (ADC,
p
= 0.826; ITSS,
p
= 0.539). After integration with ITSS, the diagnostic performance of ADC was improved for differentiating PA from WT (AUC 0.921 vs 0.873) and from MT (AUC 0.906 vs 0.882).
Conclusion
SWI could provide added information to DWI and serve as a supplementary imaging marker for the characterization of parotid gland tumors.
Objective
To assess whether the addition of salivary gland ultrasonography (SGUS) or replacement of current criteria items by SGUS influences the performance of the American College of Rheumatology ...(ACR)/European League Against Rheumatism (EULAR) criteria for primary Sjögren's syndrome.
Methods
Included were consecutive patients with complete data on all ACR/EULAR items (n = 243) who underwent SGUS in our primary Sjögren's syndrome expertise center. Clinical diagnosis by the treating physician was used as the gold standard. Separate analyses were performed for patients who underwent labial or parotid gland biopsies. The average score for hypoechogenic areas in 1 parotid and 1 submandibular gland was determined (range 0–3). Next, performance of the ACR/EULAR criteria was evaluated after addition of SGUS or replacement of current items by SGUS.
Results
Receiver operating characteristic analysis showed an optimal cutoff value of ≥1.5 for SGUS. The optimal weight for SGUS positivity was 1. Cutoff for ACR/EULAR fulfilment remained ≥4. In patients who underwent a labial gland biopsy (n = 124), the original criteria showed an area under the curve (AUC) of 0.965, sensitivity of 95.9%, and specificity of 92.2%. After the addition of SGUS, the AUC was 0.966, with a sensitivity of 97.3% and specificity of 90.2%. In patients who underwent a parotid gland biopsy (n = 198), similar results were found. Sensitivity of the criteria decreased substantially when SGUS replaced salivary gland biopsy or anti‐SSA antibodies, while performance remained equal when SGUS replaced the ocular staining score, Schirmer's test, or unstimulated whole saliva flow.
Conclusion
Validity of the ACR/EULAR criteria remains high after incorporation of SGUS. With SGUS, clinicians are offered a larger array of tests to evaluate fulfillment of the ACR/EULAR criteria.
Radiotherapy is a common treatment option for head and neck cancer patients; however, the surrounding healthy salivary glands are often incidentally irradiated during the process. As a result, ...patients often experience persistent xerostomia and hyposalivation, which deceases their quality of life. Clinically, there is currently no standard of care available to restore salivary function. Repair of epithelial wounds involves cellular proliferation and establishment of polarity in order to regenerate the tissue. This process is partially mediated by protein kinase C zeta (PKCζ), an apical polarity regulator; however, its role following radiation damage is not completely understood. Using an in vivo radiation model, we show a significant decrease in active PKCζ in irradiated murine parotid glands, which correlates with increased proliferation that is sustained through 30 days post-irradiation. Additionally, salivary glands in PKCζ null mice show increased basal proliferation which radiation treatment did not further potentiate. Radiation damage also activates Jun N-terminal kinase (JNK), a proliferation-inducing mitogen-activated protein kinase normally inhibited by PKCζ. In both a PKCζ null mouse model and in primary salivary gland cell cultures treated with a PKCζ inhibitor, there was increased JNK activity and production of downstream proliferative transcripts. Collectively, these findings provide a potential molecular link by which PKCζ suppression following radiation damage promotes JNK activation and radiation-induced compensatory proliferation in the salivary gland.
Previously (Shan et al, 2005), we reported that adenoviral vector-mediated transfer of the human aquaporin-1 (hAQP1) cDNA to minipig parotid glands following irradiation (IR) transiently restored ...salivary flow to near normal levels. This study evaluated a serotype 2, adeno-associated viral (AAV2) vector for extended correction of IR (single dose; 20 Gy)-induced, parotid salivary hypofunction in minipigs. At 16 weeks following the IR parotid salivary flow decreased by 85-90%. AAV2hAQP1 administration at week 17 transduced only duct cells and resulted in a dose-dependent increase in salivary flow to approximately 35% of pre-IR levels (to approximately 1 ml per 10 min) after 8 weeks (peak response). Administration of a control AAV2 vector or saline was without effect. Little change was observed in clinical chemistry and hematology values after AAV2hAQP1 delivery. Vector-treated animals generated high anti-AAV2 neutralizing antibody titers by week 4 (approximately 1:1600) and significant elevations in salivary (approximately 15%), but not serum, granulocyte macrophage colony-stimulating factor levels. Following vector administration, salivary Na(+) was dramatically increased, from approximately 10 to approximately 55 mM (at 4 weeks) and finally to 39 mM (8 weeks). The findings demonstrate that localized delivery of AAV2hAQP1 to IR-damaged parotid glands leads to increased fluid secretion from surviving duct cells, and may be useful in providing extended relief of salivary hypofunction in previously irradiated patients.
Pathology Mimicking Orofacial Pain Sandhu, Shaiba; Handa, Shruti
The Dental clinics of North America,
January 2023, 2023-01-00, 20230101, Letnik:
67, Številka:
1
Journal Article
Recenzirano
A case of a 64-year-old woman is reported, who developed new-onset pain over a preexisting area of right mandibular fullness. Clinical examination, MRI, and fine-needle aspiration cytology confirmed ...the diagnosis of a benign parotid gland tumor-pleomorphic adenoma, which was treated by total parotidectomy with complete removal of the tumor. When evaluating a patient with orofacial pain, oral health care providers should be cognizant of all potential differential diagnoses, especially in the setting of red flags such as persistent or enlarging facial swelling/fullness.
Objective
Previous studies have evaluated various markers as prognostic predictors in patients with many types of cancers. However, the influence of such factors on the outcomes of patients with ...parotid gland carcinoma (PGC) is unknown. This study investigated the roles of alternative markers in the prognoses of patients with PGC.
Methods
Overall, 101 patients who underwent curative treatment for PGC were retrospectively evaluated, and their 5‐year overall and disease‐free survival rates were calculated. The prognostic values of clinical and pathologic factors were determined.
Results
The 5‐year overall and disease‐free survival rates were 73.1% and 62.8%, respectively. Multivariate analysis revealed that a low lymphocyte‐to‐monocyte ratio (LMR), high T classification, high N classification, and perineural invasion were independent predictors of poor prognosis.
Conclusions
Thus, we identified LMR as an independent prognostic factor for patients with PGC. Patients with low LMRs who are amenable to treatment may require adjuvant treatment to improve their prognoses.
Level of Evidence
4 Laryngoscope, 131:E864–E869, 2021
Objective
To assess prospectively the efficacy of arterial spin labelling (ASL) against conventional and diffusion-weighted (DW) MR imaging for differentiating parotid gland tumours.
Methods
We ...included 10 pleomorphic adenomas, 12 Warthin's tumours, and nine malignant tumours of the parotid glands. Only tumours larger than 10 mm were included in this study. All parotid gland tumours underwent T1-weighted, T2-weighted, DW, and ASL imaging. Tumour-to-parotid gland signal intensity ratios (SIRs) and apparent diffusion coefficients (ADCs) of solid components were correlated with these pathologies.
Results
SIRs on T2-weighted images and ADCs were higher in pleomorphic adenomas than in Warthin's tumours (
p
< .01) and malignant tumours (
p
< .01). SIRs on ASL were higher in Warthin's tumours than in pleomorphic adenomas (
p
< .01) and malignant tumours (
p
< .05). Az value of SIRs on ASL for differentiating Warthin's tumours from the other pathologies was 0.982. The sensitivity, specificity, and accuracy of SIRs on ASL for the diagnosis of Warthin's tumours at an optimal SIR threshold of over 8.70 were 91.7 %, 94.7 %, and 93.5 %, respectively.
Conclusions
ASL with SIR measurements could non-invasively evaluate tumour blood flow of parotid gland tumours and differentiate Warthin's tumours from pleomorphic adenomas and malignant tumours.
Key Points
•
ASL non-invasively evaluates tumour blood flow of parotid gland tumours
•
ASL differentiates Warthin's tumours from pleomorphic adenomas and malignant tumours
•
ASL cannot differentiate between pleomorphic adenomas and malignant tumours
Salivary gland tumors are a rare, histologically heterogeneous group of tumors. The distinction between malignant and benign tumors of the parotid gland is clinically important. This study aims to ...develop and evaluate a deep-learning network for diagnosing parotid gland tumors
the deep learning of MR images.
Two hundred thirty-three patients with parotid gland tumors were enrolled in this study. Histology results were available for all tumors. All patients underwent MRI scans, including T1-weighted, CE-T1-weighted and T2-weighted imaging series. The parotid glands and tumors were segmented on all three MR image series by a radiologist with 10 years of clinical experience. A total of 3791 parotid gland region images were cropped from the MR images. A label (pleomorphic adenoma and Warthin tumor, malignant tumor or free of tumor), which was based on histology results, was assigned to each image. To train the deep-learning model, these data were randomly divided into a training dataset (90%, comprising 3035 MR images from 212 patients: 714 pleomorphic adenoma images, 558 Warthin tumor images, 861 malignant tumor images, and 902 images free of tumor) and a validation dataset (10%, comprising 275 images from 21 patients: 57 pleomorphic adenoma images, 36 Warthin tumor images, 93 malignant tumor images, and 89 images free of tumor). A modified ResNet model was developed to classify these images. The input images were resized to 224x224 pixels, including four channels (T1-weighted tumor images only, T2-weighted tumor images only, CE-T1-weighted tumor images only and parotid gland images). Random image flipping and contrast adjustment were used for data enhancement. The model was trained for 1200 epochs with a learning rate of 1e-6, and the Adam optimizer was implemented. It took approximately 2 hours to complete the whole training procedure. The whole program was developed with PyTorch (version 1.2).
The model accuracy with the training dataset was 92.94% (95% CI 0.91, 0.93). The micro-AUC was 0.98. The experimental results showed that the accuracy of the final algorithm in the diagnosis and staging of parotid cancer was 82.18% (95% CI 0.77, 0.86). The micro-AUC was 0.93.
The proposed model may be used to assist clinicians in the diagnosis of parotid tumors. However, future larger-scale multicenter studies are required for full validation.
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