Warthin-like papillary thyroid carcinoma: a case report and comprehensive review of the literature.

Background: Papillary Thyroid Carcinoma (PTC) is the most frequent endocrine malignancy with a variety of histological presentations. Warthin-like Papillary Thyroid Carcinoma (WLPTC) is an uncommon neoplasm that is recognized as a distinct subtype of PTC in the WHO classification of thyroid tumors. In this report, we present a novel case of WLPTC in a female patient and provide an in-depth review of the available literature on its clinical, pathological, and therapeutic characteristics. Case presentation: A 27-year-old female patient was referred for neck swelling. Ultrasound showed two suspicious thyroid nodules leading to a thyroidectomy. She was diagnosed with intermediate-risk bifocal foci of classic PTC and WLPTC, arising from a background of chronic lymphocytic thyroiditis (CLT). This pT1b(m) N1b M0 malignancy was treated with adjuvant isotopic ablation and suppressive thyroxine therapy. The 1-year outcomes were favorable. Literature review: It covered articles published from 1995 to 2022, by searching PubMed and Google Scholar using specific terms. Out of 148 articles reviewed by two authors, 25 relevant articles were selected, including 13 case reports and 12 case series. The study included 150 cases of WLPTC. Data related to clinical presentation, imaging, histological features, management, and outcomes, were extracted. The mean age of diagnosis was 39 years, with a female predominance. The most common clinical presentation was neck swelling. Thyroid autoimmunity was positive in 71.6% of patients. Lymph node metastases were present in 28% of cases, with no reported distant metastases. Overall, the outcomes were favorable. Conclusion: WLPTC shares similar clinical and radiological presentations as classic PTC. The hallmark histological features of WLPTC are papillae lined with oncocytic tumor cells with papillary nuclear changes and lymphoid stroma. WLPTC is almost constantly associated with CLT. The management of WLPTC aligns with that of classic PTC with comparable stage and risk category, often resulting in favorable outcomes.

A Case of Intra-Amniotic Umbilical Vein Varices Misdiagnosed as an Omphalocele.

Background: Intra-amniotic umbilical vein varices are characterized by a focal dilatation of the extra abdominal umbilical vein. Case report: We report a full-term baby female with extra-abdominal umbilical vein varices misdiagnosed clinically as an omphalocele. The umbilical vein was ligated and excised near the level of the liver. The infant died one day after surgery due to extrinsic compression of the renal pedicle by a massive thrombus, resulting in severe renal failure and life-threatening hyperkalemia despite intensive resuscitation. Conclusion: Large intra-amniotic umbilical vein varices can be clinically misdiagnosed as an omphalocele. Their resection near the level of the fascia, as with normal umbilical veins, could be a better management with a better prognosis.

Phyllodes Tumor of the Bladder in a 2-Year-Old Boy - An Exceptional Finding.

Background: Breast phyllodes tumor has a distinct histologic appearance. There are no pediatric phyllodes tumors of the bladder in English literature reported. Case report: A 2-year-old boy presented with a urinary infection and obstructive urinary symptoms. A 3-cm slow-growing bladder mass revealed by repeated transabdominal ultrasonography was initially considered a ureterocele. Cystoscopic and laparoscopic exploration using pneumovesicum confirmed the diagnosis of a bladder neck tumor. Histologically, the features were of a benign phyllodes tumor, morphologically similar to those seen in breast tissue. The patient received no further treatment and showed no recurrence or metastasis. Conclusion: Phyllodes tumor can cause a pediatric bladder tumor.

Immunohistochemical Overexpression of Cyclin D1 in Tunisian Invasive Breast Carcinoma Women.

BACKGROUND: Breast cancer represents the most frequent cancer and cause of death in women worldwide and in Tunisia. Cyclin D1 is a gene of cell cycle regulation. It represents a potential oncogene in invasive breast cancer; however; the results are conflicting. We performed a retrospective study aiming to analyze the prognostic impact of cyclin D1 expression in patients with invasive breast carcinoma of no special type and its relation with clinical-pathological features. METHODS: One hundred cases of invasive breast carcinoma of no special type diagnosed between 2009 and 2011 were included in this study. Immunohistochemical (IHC) staining was performed for cyclin D1 in all cases. Results were analyzed statistically. RESULTS: Cyclin D1 positivity was seen in 74 cases (74%), of which 32 cases (32%) showed strong immunoreactivity. Cyclin D1 staining was statistically significantly associated with estrogen receptor (ER) and progesterone receptor (PR) positivity (P<0.0001) and with low grade SBR (P=0.007). None of the clinical data and other pathological features had any association with cyclin D1 expression (P>0.05). Univariate analysis revealed that expression of cyclin D1 was not statistically associated with overall survival (OS) and disease-free survival (DFS) (P=0.459 and P=0.564, respectively). CONCLUSION: These results confirm that cyclin D1 overexpression can be employed as a beneficial prognostic marker and suggest that anti-cyclin D1 therapy may be efficient, especially for ER positive tumors.

Atypical spindle cell lipomatous tumor of the tongue: A rare entity arising in an unusual location.

Atypical spindle cell/pleomorphic lipomatous tumor (ASCPT) constitutes an emerging entity of lipomatous tumors. It is a benign tumor. It occurs typically in limbs and limb girdles. The occurrence in oral cavity is unusual. The diagnosis of this neoplasm is challenging. Herein, we report a case of ASCPT arising in the tongue.

Alveolar Adenoma: A Rare Benign Tumour of the Lung with A Challenging Diagnosis.

Alveolar adenoma is a rare lung benign tumour originating from type II pneumocytes. It presents as a well-defined nodule. In some cases, it is difficult to differentiate from lung cancer. Few cases of this tumour have been reported. We describe here a case of alveolar adenoma in a 63-year-old man discovered incidentally on chest X-ray. The lesion was reported as lepidic adenocarcinoma in bronchoscopic biopsy. The patient underwent a thoracoscopic left lower lobectomy. The histopathological and immunohistochemical examinations resulted in a diagnosis of alveolar adenoma. We report this case to describe its morphological and immunohistochemical characteristics and to emphasize its diagnostic difficulties.

Immune checkpoint molecules B7-H6 and PD-L1 co-pattern the tumor inflammatory microenvironment in human breast cancer.

B7-H6 and PD-L1 belong to the B7 family co-stimulatory molecules fine-tuning the immune response. The present work investigates the clinical effect of B7-H6 protein expression with PD-L1 status and the infiltration of natural killer cells as potential biomarkers in breast tumor inflammatory microenvironment. The expression levels of B7-H6 protein by cancer cells and immune infiltrating cells in human breast cancer tissues and evaluate their associations with PD-L1 expression, NK cell status, clinical pathological features and prognosis were explored. The immunohistochemistry labeling method was used to assess B7-H6 and PD-L1 proteins expression by cancer and immune cells. The associations between immune checkpoint, major clinical pathological variables and survival rates were analyzed. B7-H6 protein was depicted in both breast and immune cells. Results showed that Tumor B7-H6 expression is highly associated with Her-2 over expression. B7-H6 + immune cells are highly related to the Scarff-Bloom-Richardson grade and associated with PD-L1 expression and NK cells status. Survival analysis revealed a better prognosis in patients with low expression of B7-H6 by cancer cells. Conversely, B7-H6 + immune cells were significantly associated with longer survival. Findings strongly suggest an interaction between B7 molecules that contributes to a particular design of the inflammatory microenvironment. This may influence the efficiency of therapies based on antibodies blocking the PD-L1/PD1 pathway and can explain the detection of clinical benefits only in a fraction of patients treated with immune checkpoint inhibitors.

Prognostic Value of Natural Killer Cells Besides Tumor-Infiltrating Lymphocytes in Breast Cancer Tissues.

BACKGROUND: Each subgroup of immune cells has a different prognostic role in breast cancer; however, the prognostic impact of tumor-infiltrating natural killer cells (TINKs) is still not well established. Our aim was to assess the prognostic impact of natural killer (NK) cells in breast carcinomas. MATERIALS AND METHODS: NK cells infiltration were assessed by immunohistochemistry (IHC). Statistical analyses were performed to evaluate the correlation of NK cells with clinical-pathological features and outcome. RESULTS: CD56 IHC was realized in 126 patients. NK cells infiltration showed significant and positive association with tumor high Scarff-Bloom-Richardson (SBR) grade. NK cells were significantly associated with HER2-positive breast cancer and triple-negative breast cancer subtypes. Analyses showed significant and inverse correlation with progesterone and estrogen receptors expression status. High NK cells were significantly related to high Ki-67 labeling index. Our data showed that high NK cells infiltrate was significantly associated with tumor-infiltrating lymphocytes in breast cancer tissues. At a median follow-up of 5.5 years, high CD56 expression (≥ 5 cells/10 high power field) was associated significantly with a good overall survival and with good disease-free survival. CONCLUSION: In this study, we assessed the important prognostic role of TINKs in breast carcinomas, which seems to be evident despite its association with aggressive pathological features. Thus evaluation of NK cells can be standardized and integrated in daily routine.

An uncommon breast metastasis of mediastinal neuroblastoma within a child: A case report.

A 13-year-old girl presented with dull lumbar pain and recent paraplegia. Clinical examination found a breast mass associated with an axillary adenopathy. Imaging revealed a large mass in the posterior mediastinum with spinal cord compression. Histological analysis confirmed the diagnosis of neuroblastoma with mammary metastases. To the best of our knowledge, breast metastasis is being reported for the first time in a mediastinal neuroblastoma.

FOXA1 Expression in Nasopharyngeal Carcinoma: Association with Clinicopathological Characteristics and EMT Markers.

The forkhead box (FOXA) family of transcription factors regulates gene expression and chromatin structure during tumorigenesis and embryonic development. Until now, the relationship between FOXA1 and the nasopharyngeal carcinoma (NPC) has not yet been reported. Therefore, our purpose is to analyze the expression of FOXA1 in 56 NPC patients compared to 10 normal nasopharyngeal mucosae and to correlate the expression with the clinicopathological features. Besides, we investigated the association between FOXA1 and LMP1 gene expression, as well as the EMT markers namely the E-cadherin and Twist1. Among 56 NPC tissues, 34 (60.7%) cases were positive for FOXA1. Furthermore, we noticed that FOXA1 expression correlated with TNM (p = 0.037), and age at diagnosis (p = 0.05). Moreover, positive expression of FOXA1 is likely to be associated with prolonged disease-free survival and overall survival rates. On the other hand, we observed a positive association between the expression of E-cadherin and FOXA1 (p = 0.0051) whereas Twist1 correlated negatively with FOXA1 (p = 0.004). Furthermore, knowing that LMP1 plays a key role in the pathogenesis of NPC, we explored the association of FOXA1 with the LMP1 gene expression in both NPC cell lines and tissues. We found that, in the C666-1 which displays low levels of LMP1, the expression of FOXA1 is high, and inversely in the C15 cell line that expresses a high level of LMP1, the level of FOXA1 is low. Besides, in accordance to our results, we found that in NPC tissues there is a negative association between LMP1 and FOXA1. In conclusion, our results suggest that the overexpression of FOXA1 is associated with a nonaggressive behavior and favorable prognosis in NPC patients. FOXA1 could contribute in the EMT process through key factors as E-cadherin, Twist1, and LMP1.

Renal angiomyolipoma: Clinico-pathologic study of 17 cases with emphasis on the epithelioid histology and p53 gene abnormalities.

BACKGROUND: Epithelioid angiomyolipoma (EAML) is a rare potentially malignant variant of renal angiomyolipoma (RAML). This study aims to determine whether RAML clinico-pathologic and molecular features (i.e. p53 gene abnormalities) differ significantly with regards to its histologic variant or to the presence of an epithelioid component within it. METHODS: Consecutively resected RAML were reviewed, tumours comprising at least 80% of epithelioid cells were considered as EAML according to the 2016 World Health Organization classification of tumours of the kidney. P53 gene abnormalities were investigated using both immunohistochemical and molecular analysis. RESULTS: A total of 3 EAML among 17 RAML were identified, accounting for 3.9% of the total AML cases. Fatty aspect on imaging was more observed within tumours devoid of an epithelioid component. EAML showed a higher mitotic rate and a stronger p53 staining, no renal poles involvement and was not treated by nephron sparing surgeries. RAML comprising an epithelioid component demonstrated severer nuclear atypia as well as stronger p53 staining. P53 gene sequencing revealed a missense mutation (c.747G > C) in one classic AML harbouring a strong labelling with p53. CONCLUSIONS: Strong p53 staining in a RAML, even in the absence of gene mutation, may suggest the presence of an epithelioid component or of a truly EAML. To the best of our knowledge, c.747G > C p53 gene mutation is being reported for the first time in a RAML, although its role in AML pathogenesis is still unknown.

Prognostic Role of Lymphovascular Invasion in Patients with Urothelial Carcinoma of the Upper Urinary Tract.

PURPOSE: To evaluate the impact of lymphovascular invasion on the prognosis of patients treated for upper urinary tract urothelial carcinomas. MATERIALS AND METHODS: Clinical records of 49 patients treated surgically at our institute for upper urinary tract urothelial carcinomas were reviewed retrospectively. LVI was defined as the presence of cancer cells within an endotheluim-lined space without underlying muscular walls. Actuarial survival curves were analysed by Kaplan-Meier method. Multivariate analysis was performed using Cox's proportional hazard model. RESULTS: Median follow-up was 32 months. Lymphovascular invasion was present in 26 (53%) patients. Lymphovascular invasion was associated with higher pathological tumor stage (pT) and higher tumor grade. The disease-free and overall survival rates of the patients with lymphovascular invasion were significantly worse than those of the patients without lymphovascular invasion (p < 0.001 and p = 0.027 respectively). Multivariate analysis revealed that lymphovascular invasion as well as tumor grade and pathological tumor stage were significant prognosticfactors for disease-free and overall survival. CONCLUSION: The presence of lymphovascular invasion was a strong predictor of a poor outcome for UTUC. This finding could help identify patients at greater risk for disease recurrence who would benefit from close follow-up and early adjuvant therapy.

Plasmacytoid melanoma of the urinary bladder and lymph nodes with immunohistochemical expression of plasma cell markers revealing primary esophageal melanoma.

Plasmacytoid variant of melanoma is reported in only rare cases. We present the case of a 54-years-old man admitted for enlarged lymph nodes in the lumbar region. Initial diagnosis of plasmablastic lymphoma/plasma cell myeloma was considered. At our institute, a bladder polyp was removed. Microscopic exam demonstrated dense plasmacytoid cells infiltration with pigment deposits. Immunohistochemical study showed strong expression of HMB45, Melan A, and vimentin. There was focal positivity with S100 protein and CD138/syndecan-1. The diagnosis of metastatic plasmacytoid melanoma was finally established. Clinical exam revealed an esophageal melanoma with melanosis supporting its primary location. Although rarely, melanoma especially plasmacytoid variant may express plasma cell markers which may lead to erroneous diagnosis of plasma cell proliferation. Careful morphological examination for melanin pigment and the use of panel of melanocytic markers are helpful for diagnosis.

Mullerian adenosaroma of the cervix with sarcomatous overgrowth and heterologous elements presenting as a recurrent cervical polyp.

Mullerian adenosarcoma of the cervix is a rare tumor composed of benign epithelial and malignant stromal components. Sarcomatous overgrowth and heterologous elements in cervical adenosarcoma are extremely infrequent. We report the case of a 26-year-old woman admitted at the gynaecology department for a painless mass protruding from her vagina. The initial pathological exam concluded to endocervical polyp. Six months later, the patient was readmitted with a recurrence of the polyp. The pathological exam demonstrated interlacing fascicles of elongated spindle cells with few mitotic activity and no glandular formation. After reviewing of the initial polyp the diagnosis of mullerian adenosarcoma was suggested. A second recurrence of the polyp was noted one month later. Histopathological exam of the recurrent polyp confirmed the diagnosis of adenosarcoma with sarcomatous overgrowth and heterologous elements. The patient was lost for follow-up. Cervical adenosarcoma with sarcomatous overgrowth and heterologous element is a rare tumor that occurs in younger age in contrast to endometrium/corpus uterin mullerian adenosarcoma. In young women with recurrent cervical polyp, mullerian adenosarcoma must be considered and should be excluded by careful histopathological exam. Sarcomatous overgrowth and myometrial invasion are the most important prognostic factors. Treatment strategy is still unclear.

[Abrikosoff's tumor: a clinicopathologic study of nine cases]. / Tumeur d'Abrikosoff : etude anatomoclinique de neuf cas.

BACKGROUND: Abrikossoff's tumour or granular cell tumor or is a benign neurogenic tumour. It is ubiquitous with the most frequently affected site is the head and neck region. AIMS: To report a series of granular cell tumors and to discuss its clinicopathologic features and histogenesis. METHODS: We report a series of nine cases diagnosed between January 2004 and December 2006 in the Pathology Department of the University Hospital of Sfax. We have collected the clinical aspects and we have proceeded on a pathological, cytochemical (PAS and PAS diastasis) and immunohistochemical study. RESULTS: Nine cases of TCG are presented: 5 females and 4 males. The median age was 33.9 years (extremes: 7 and 53 years). All tumours were unique. The most common localization was in the head and neck region (5 cases). The diagnosis was suspected using standard histologic criteria and confirmed by immunohistochemistry: tumour cells expressed vimentine (90%), S100 protein (100%) and neuron specifique enolase (80%). In all cases patients were treated by excisional resection and had a benign course with no evidence of recurrence (median follow up: 2 years). CONCLUSION: Granular cell tumours are rare neoplasm which must be recognised because they demonstrate a benign behaviour after their surgical excision. Histological features of granular cell tumours are commonly characteristic but some times they can be misdiagnosed as malignant tumours especially when the biopsy is superficial. The staining for neurogen markers and PAS are useful tools.

Study of the Bacillus thuringiensis Vip3Aa16 histopathological effects and determination of its putative binding proteins in the midgut of Spodoptera littoralis.

The bacterium Bacillus thuringiensis produces, at the vegetative stage of its growth, Vip3A proteins with activity against a broad spectrum of lepidopteran insects. The Egyptian cotton leaf worm (Spodoptera littoralis) is an important agricultural pest that is susceptible to the Vip3Aa16 protein of Bacillus thuringiensis kurstaki strain BUPM95. The midgut histopathology of Vip3Aa fed larvae showed vacuolization of the cytoplasm, brush border membrane destruction, vesicle formation in the apical region and cellular disintegration. Biotinylated Vip3Aa toxin bound proteins of 55- and 100-kDa on blots of S. littoralis brush border membrane preparations. These binding proteins differ in molecular size from those recognized by Cry1C, one of the very few Cry proteins active against the polyphagous S. littoralis. This result supports the use of Vip3Aa16 proteins as insecticidal agent, especially in case of Cry-resistance management.

[Ewing sarcoma osseous and extraosseous : a clinicopathologic study of 29 cases]. / Sarcome d'Ewing Osseux et Extra Osseux : Etude anatomo-clinique de 29 cas.

BACKGROUND: Ewing's sarcoma (ES) is a rare tumour accounting for 10% of primary malignant bone tumours in children and 3% of all childhood malignancies. ES belongs to a group of small round cell tumours. AIM: In this review, we will describe the main clinicopathological features of this rare tumour and discuss its prognosis. METHODS: We report a retrospective study of 29 cases of ES, of which 4 were extraosseous, diagnosed over a period 11 years (January 1989 - December 1999). Clinicopathological data were described. Hematoxylin-eosin staining and immunohistochemical study were reviewed. RESULTS: 12 patients were male and 17 were female (ratio: 0,8) with a median age of 16 years. 62,5% of tumours were located in flat bone and 33,3% in long bone. The medium size of the tumor was 10,6 cm (range:3-25cm). 27,5% of patients presented with metastatic disease at time of diagnosis. Microscopically, tumour tissue was composed of round, small, blue cells with fine granular chromatin. Tumour cells strongly coexpressed CD99 and vimentin (100%). Systemic treatment consisted of adjuvant chemotherapy (84,2%). Local control was based on and surgery (57,9%) or radiation therapy (36,8%). A good response to chemotherapy was obtained in 37.5%; 13,7% of patients were alive without disease (medium follow up: 169 mois); 34,5% of patients developed metastases (medium follow up : 23 months) and 10,3% developed recurrences (medium follow up :13 months). CONCLUSION: Our study emphasizes two points : the great size of the tumor and the frequent location in flat bone which may explain the poor prognosis of Ewing sarcoma in our series despite the multidisciplinary treatment.