Juvenile Papillomatosis of the Breast: a Report of Two Cases with Review of the literature.

Background: Juvenile papillomatosis (JP) of the breast is a rare and benign proliferative disorder affecting young women. The affected patients tend to have an increased risk of breast cancer development during follow-up. Objective: This article aims to highlight a rare entity of breast disease, that harbor risk of breast cancer. Case Presentation: Here, we present 2 cases of JP in young females; the first case is a 13 year-old presented with spontaneous nipple discharge, while the other patient is a 24 year-old presented with a right breast lump. Both patients had a total excision of the breast lesions, revealing JP at histology. Discussion: Juvenile Papillomatosis is considered a clinicopathological entity and is usually misdiagnosed as fibroadenoma clinically and radiologically, which requires histological correlation. The histologic findings are well-defined (hyperplasia, papillomatosis, and multiple cysts with foamy histiocytes).The controversy in management between surgery and observation is because of insufficient knowledge about the direct relationship between JP and subsequent cancer. Conclusion: Considering the risk of developing breast cancer in JP, enrolling patients and their families in a close follow-up and surveillance program is crucial.

Challenging diagnosis of male intraductal papilloma masquerading as eccrine hidradenoma in the breast: Case report.

RATIONALE: This article presents a challenging case involving an elderly male patient with a misdiagnosed intraductal mammary papilloma initially identified as a sweat adenoma through ultrasound imaging. The study aims to explore the histopathology, clinical presentations, and sonographic features of both conditions, emphasizing the contributing factors to the diagnostic misstep. PATIENT CONCERNS: A 61-year-old male reported a persistent left breast mass, along with pain and swelling, spanning a 6-month duration. DIAGNOSES: Ultrasound examination indicated a deep, square, mixed-echo mass in the left nipple, initially suggestive of a sweat adenoma. However, subsequent pathological analysis following resection under general anesthesia confirmed an intraductal papilloma. INTERVENTION: The patient underwent surgical resection of the left breast mass under general anesthesia. OUTCOME: Post-surgery, the patient exhibited satisfactory recovery; however, regrettably, he was lost to follow-up. LESSONS: This study underscores the challenge in differentiating between clear cell sweat adenoma and male intraductal mammary papilloma solely based on ultrasonic characteristics. It emphasizes the susceptibility of ultrasound-based diagnoses to misinterpretation, highlighting the critical need for a comprehensive pathological examination to establish a definitive diagnosis.

[Food hygiene and safety - What is your diagnosis?] / Lebensmittelhygiene ­ Was ist Ihre Diagnose?

INTRODUCTION: A healthy, 1,5 year old female wild boar (Sus scrofa) was shoot in a hunting district in Switzerland on June 22, 2023. The meat inspection revealed noticeable skin changes on all four distal extremities which were described histologically as multifocal epidermal hyperplasia and moderate orthokeratotic hyperkeratosis. The rest of the animal body, the organs and the lymph nodes were without any obvious lesions. The diagnosis of papilloma virus-associated exophytically growing papillomas was made. The venison was approved as fit for human consumption.

INTRODUCTION: Un sanglier femelle (Sus scrofa) en bonne santé, âgé d'un an et demi, a été abattu dans le nord de la Suisse le 22 juin 2023. L'inspection de la viande a révélé des modifications cutanées notables sur les quatre extrémités distales des membres qui ont été décrites histologiquement comme une hyperplasie épidermique multifocale et une hyperkératose orthokératosique modérée. Le reste du corps de l'animal, les organes et les ganglions lymphatiques ne présentaient aucune lésion évidente. Le diagnostic de papillomes à croissance exophytique associés à des papillomavirus a été posé. La venaison a été déclarée propre à la consommation humaine.

Clinical characteristics of intradermal nevus in the external auditory canal.

Melanocytic nevus occurring in the external auditory canal (EAC) is uncommon. Therefore, in the absence of sufficient clinical experience, this disease may be challenging to be suspected even with a physical examination. Herein, clinical and demographic features of intradermal nevus in the EAC were investigated. Patients with an intradermal nevus on histopathological examination (n = 15; 2 men and 13 women; P = .005) were included from a pool of patients who underwent surgical resection for EAC tumors between November 2011 and March 2022. Data were retrospectively collected on their sex, appearance of the lesions, surgical method, and outcomes. Nine patients had a dome shape and 6 patients had a papillomatous shape. Ten patients had dark colors and 5 patients had pale colors. All pale colors appeared only in the dome-shaped nevus, and only dark colored papillomatous nevi were observed. A significant difference was noted in terms of the frequency of occurrence by colors (dark or pale) according to shape (dome or papillomatous) (P = .044). No patient showed recurrence or EAC stenosis after circumferential excision. In women, a dome-shaped or papillomatous mass with dark color in the EAC may likely be predicted as the nevus. However, it can also occur in men, and even if it is a pale color, in the case of a dome shape, taking the nevus into account is important. Moreover, successful treatment outcomes were achieved through circumferential excision.

Prevalence and interpretation of AFF immunostain of DEK::AFF2 fusion-associated papillary squamous cell carcinoma in a retrospective cohort of recurrent sinonasal papillomas.

DEK::AFF2 fusion-associated papillary squamous cell carcinoma is a novel entity characterized by its unique translocation and malignant clinical course. In this study, AFF immunohistochemistry (IHC) was performed in recurrent sinonasal papillomas for reviewing the prevalence of undiagnosed DEK::AFF2 carcinomas and to investigate the performance of AFF IHC in diagnosis of DEK::AFF2 carcinomas. Recurrent sinonasal papillomas after surgical excision in a two-decade period were retrieved. Histologic slides were reviewed for features of DEK::AFF2 carcinoma. AFF IHC was performed, and cases with any (> 1%) nuclear positivity were validated by DEK break apart fluorescence in situ hybridization. Totally 43 cases were included, comprising 28 inverted, 6 exophytic, one oncocytic, and 8 non-specified sinonasal papillomas. Five (11.6%) cases exhibited positivity to AFF IHC. Three cases exhibited patchy weak to moderate staining intensity predominantly in a granular cytoplasmic pattern. Two cases exhibited strong and diffuse (> 90%) nuclear staining. Cases showing weak staining were negative for DEK rearrangement, while those with strong staining were positive. Both cases of DEK::AFF2 carcinoma showed aggressive behavior with extensive local invasion and nodal metastasis. Background stromal plasma cells, when present, consistently showed strong and diffuse staining. AFF IHC was further performed in plasmacytoma samples as control and showed strong and diffuse immunoreactivity. A significant minority of recurrent sinonasal papillomas represent DEK::AFF2 carcinomas. Granular, cytoplasmic, or incomplete AFF staining should be considered as negative. In view of the rarity of DEK::AFF2 carcinomas, plasma cells and plasma cell neoplasms are potential for internal and surrogate external controls.

Ultrasound imaging of onychopapilloma: Evaluation of 43 cases.

INTRODUCTION: Onychopapilloma is a benign tumor presenting as an isolated benign warty distal matrix and bed lesion in association with longitudinal erythronychia for which no underlying cause can be identified. Diagnosis might be challenging due to other, more severe, causes of erythronychia. OBJECTIVES: This study aimed to investigate the clinical and sonographic features of onychopapilloma using high-frequency ultrasound. METHODS: Between February 2022 and May 2023, a cross-sectional study was conducted, involving 39 patients displaying clinical features suggestive of onychopapilloma. These patients were selected at random and underwent examination using high-frequency ultrasound. High-frequency ultrasound assessments of the nail unit were carried out using 33 MHz and 24 MHz high-frequency linear transducers. An experienced radiologist performed the ultrasound scans while participants assumed an orthostatic seated position. Subsequently, the collected ultrasound images were independently evaluated by two radiologists specializing in nail injuries, ensuring a blinded assessment. This evaluation aimed to provide comprehensive insights into the imaging characteristics of onychopapilloma. RESULTS: High-frequency ultrasound revealed oval or linear hyperechoic masses located in the middle of the nail plate, without vascularity or posterior acoustic shadowing. Nail plate irregularity, thickening, and nail bed indentation were observed in some cases. These findings demonstrate that high-frequency ultrasound can aid in the diagnosis and surgical planning of onychopapilloma, providing detailed visualization of the lesion and surrounding structures. CONCLUSIONS: This imaging method could represent a potential tool to differentiate onychopapilloma from other nail conditions, especially malignant neoplasms. However, further research is needed to validate these findings and assess long-term outcomes.

Ultrasound Features of Onychopapilloma at High-Frequency and Ultra-High Frequency.

OBJECTIVE: This study aimed to identify the sonographic features of pathologically confirmed onychopapilloma cases. METHODS: High-frequency up to 24 MHz and ultra-high frequency-ultrasound up to 71 MHz examinations were performed and correlated with their clinical and pathologic presentations. RESULTS: Twenty-two cases met the criteria. Clinical presentations revealed longitudinal erythronychia in 63.3% of cases. The ultrasound examinations identified a hypoechoic band in the nail bed (86.3%), nail plate abnormalities including upward displacement (68.2%) and thickening (68.1%), focal hyperechoic focal spots on the nail plate (50%) and irregularities of the ventral plate (33.3%). Color Doppler imaging showed no hypervascularity of the nail bed in all studies. These findings correlate with histological characteristics of onychopapilloma, including nail bed acanthosis, papillomatosis, and layered hyperkeratosis. Recurrence occurred in two cases after surgery, with tumors showing proximal extension in the matrix region on ultrasound not evident during clinical examination. CONCLUSION: High-frequency and ultra-high-frequency can provide anatomical information in onychopapilloma that could enhance understanding and management.

Swiss cheese disease: A rare case of juvenile papillomatosis of the breast mimicking carcinoma in a young female.

Juvenile papillomatosis is a rare benign proliferative lesion of breast seen in young females. These patients have been reported to have a strong family history for carcinoma breast. It is rarely diagnosed preoperatively but has distinct histopathological features on postoperative examination. The subsequent treatment and prognosis vary accordingly. Here, we report a case of a 38-year-old female who was operated with a preliminary diagnosis of Mucinous carcinoma of left breast.

Erosive lesion of the nipple: What is your diagnosis?

Erosive and oozing lesions of the nipple, especially when unilateral, should raise suspicion about malignant neoplasms. Herein we report a patient with typical clinical and histopathological features of erosive adenomatosis of the nipple (EAN). It is an uncommon, benign proliferative process of lactiferous ducts of the nipple. Clinically, EAN is characterized by erosion, serous discharge, edema, itching, and erythema of one of the nipples. Complete excision of the tumor yields excellent results. Although EAN is a rare disease, clinicians must be aware of this benign neoplasm in patients with erosive lesions of the nipple. The main concern is the need to rule out malignant proliferations in the differential diagnosis. Histology is the gold standard for diagnosis. The coexistence of nipple adenoma and breast cancer is well-reported in the literature. Thus, it is necessary to encourage patients with a history of EAN to maintain regular breast screening.

The First Report of Cytology of DEK::AFF2 Fusion-Associated Papillary Squamous Cell Carcinoma on Fine-Needle Aspiration.

DEK::AFF2 fusion-associated papillary squamous cell carcinoma is a recently characterized sinonasal malignancy defined by its unique translocation. DEK::AFF2 carcinomas may be deceptively monotonous and lack keratinization, resembling transitional epithelium. The lack of traditional cytological atypia presents diagnostic challenges. Our case describes the first report of fine-needle aspiration cytology of a lymph node involved by DEK::AFF2 carcinoma in a patient with previously resected sinonasal inverted papilloma with carcinomatous transformation six years prior to presentation. This aspirate consisted of a lymphoid-rich background admixed with a moderate amount of epithelial cells arranged in cohesive structures of variable size, including large sheets. The tumor cells resembled those of the corresponding biopsy, featuring mildly hyperchromatic nuclei with fine to vesicular chromatin. Lesional cells lacked keratinization, mitoses, or hyperchromasia. Our finding suggests that in nodal aspirates of patients with a history of sinonasal-type papillomas, especially those with prior malignant transformation or atypia, there should be consideration for the possibility of DEK::AFF2-related primary. When in doubt, DEK FISH of AFF2 immunohistochemistry should be performed for confirmation.

The tumor microenvironment state associates with response to HPV therapeutic vaccination in patients with respiratory papillomatosis.

Recurrent respiratory papillomatosis (RRP) is a rare, debilitating neoplastic disorder caused by chronic infection with human papillomavirus (HPV) type 6 or 11 and characterized by growth of papillomas in the upper aerodigestive tract. There is no approved medical therapy, and patients require repeated debulking procedures to maintain voice and airway function. PRGN-2012 is a gorilla adenovirus immune-therapeutic capable of enhancing HPV 6/11-specific T cell immunity. This first-in-human, phase 1 study (NCT04724980) of adjuvant PRGN-2012 treatment in adult patients with severe, aggressive RRP demonstrates the overall safety and clinically meaningful benefit observed with PRGN-2012, with a 50% complete response rate in patients treated at the highest dose. Responders demonstrate greater expansion of peripheral HPV-specific T cells compared with nonresponders. Additional correlative studies identify an association between reduced baseline papilloma HPV gene expression, greater interferon responses and expression of CXCL9 and CXCL10, and greater papilloma T cell infiltration in responders. Conversely, nonresponders were characterized by greater HPV and CXCL8 gene expression, increased neutrophilic cell infiltration, and reduced T cell papilloma infiltration. These results suggest that papilloma HPV gene expression may regulate interferon signaling and chemokine expression profiles within the tumor microenvironment that cooperate to govern clinical response to therapeutic HPV vaccination in patients with respiratory papillomatosis.

[Clinicopathological analysis of benign mammary ductal cystic papillomatosis with loss of myoepithelial cells].

Objective: To investigate the histopathological and immunohistochemical characteristics of benign apocrine cystic papillary hyperplasia of the breast with loss of myoepithelial cell layer. Methods: The clinical data, histopathological features and immunohistochemical profile of patients with benign apocrine cystic papillary hyperplasia of breast with loss of myoepithelial cell layer from January 2016 to December 2021 were examined, in which six patients were identified. Results: All six patients were female, aged 36-61 years (median 46 years), who presented with a breast mass; three cases were from the left breast and three cases were from the right breast. Microscopic examination of all cases showed breast hyperplasia with apocrine cysts, accompanied by different degrees of micropapillary and papillary hyperplasia of apocrine cells. One case was associated with lobular carcinoma in situ, and one case was associated with apocrine ductal carcinoma in situ with intraductal dissemination in adenosis. Immunohistochemical staining of CK5/6, p63, SMA, SMMHC, Calponin and CD10 showed complete absence of myoepithelial cell layer surrounding ducts in apocrine cystic papillary hyperplasia. Conclusions: The myoepithelial cells of apocrine cystic papillary hyperplasia of the breast may undergo abnormal changes and may even be completely lost. The diagnosis should be comprehensively considered along with cytomorphological and histological features to avoid overdiagnosis.

A rare case: Endobronchial solitary mixed papilloma.

Endobronchial solitary papillomas are extremely rare lung neoplasms originating from the bronchial surface epithelium. They often present with cough or recurrent hemoptysis. These tumors are benign, but they should be followed closely because they may even have a low probability of malignant transformation features. It should be kept in mind that malignancy may develop especially if the patient is a smoker. Although the etiology is not known for certain, it is thought to be caused by human papillomavirus in some cases. A 43-year-old male patient was admitted with a complaint of chronic cough. Rigid bronchoscopy was performed for diagnostic and therapeutic purposes after imaging techniques revealed a lesion obstructing the lumen of the right main bronchus. The pathology result was reported as mixed bronchial papilloma. We aimed to present our case because of its rarity and to indicate that chronic cough must be further evaluated.

Use of extracorporeal membrane oxygenation during resection of recurrent laryngeal papillomatosis: A case report.

RATIONALE: Recurrent laryngeal papillomatosis (RLP) is a common benign tumor in the larynx of children, which is characterized by high recurrence rate and rapid growth, leading to clinical symptoms such as hoarseness and difficulty breathing. Low-temperature plasma radiofrequency ablation (RFA) is the main treatment, but ventilation problems are often encountered during surgery. PATIENT CONCERNS: We report a case of a 2-year-old child with RLP who underwent low-temperature plasma RFA with the assistance of a laryngoscope. However, the surgery had to be temporarily stopped due to ventilation difficulties and difficulty in maintaining blood oxygen saturation during the procedure. DIAGNOSIS: The child was diagnosed with RLP. INTERVENTIONS: The child underwent low-temperature plasma RFA supported by laryngoscopy assisted by ECMO. OUTCOMES: Despite ventilation problems during surgery, the use of ECMO support helped maintain good oxygen saturation in the child and provided a clear surgical field, enabling the tumor to be quickly and cleanly removed. Therefore, the use of ECMO provided critical support during the surgery. LESSONS: This case highlights the importance of airway management during laryngeal papillomatosis surgery. A thorough airway assessment should be performed before anesthesia, and early use of ECMO can reduce harm to the child and ensure the child's safety.

Diagnosis and phylogenetic analysis of bovine papillomaviruses in cattle papillomatosis cases by different methods.

Papillomaviruses, known as epitheliotropic, cause proliferation in the skin, mucosa, and different visceral organs. In this study, it was aimed to diagnose bovine papillomavirus (BPV) by using different methods in the lesion taken from twenty cattle with papillomas in different areas of the body and to reveal its molecular characterization. In our study, molecular, immunohistochemistry, and transmission electron microscopy (TEM) methods were used for virus identification. Additionally, sequencing analysis was used to ascertain the phylogenetic relationship between the obtained field strains and other isolates submitted to GenBank. Histopathological analyses of the collected samples were done in addition to diagnostic procedures. Intranuclear virus particles were detected when the papillomas were investigated with TEM. In PCR analyses using degenerate and type-specific primer sets, the presence of BPV nucleic acid was determined in 70% (14/20) and 90% (18/20) of the samples, respectively. No virus could be detected in PCR applications using MY 09/11 degenerate primer sets. Twenty animals of different ages, races, and genders included in the study by random sampling method from different herds were divided into 4 groups according to the body regions where the lesions were located. Sequence analysis was performed on a sample from each group that showed strong positivity in the PCR technique using FAP 59/64 degenerate primer set and type-specific primer set. Sequence analyses were performed using FAP 59/64 degenerate primers of amplicons for phylogenetic research. In these analyses, three of the isolated strains were identified as BPV-1, which is in the Deltapapillomavirus 4 genus, and one as BPV-2. As a result of the study, it was concluded that molecular and phylogenetic studies using type-specific primers are more beneficial in order to fully reveal the etiology of papillomatosis in cattle and it would be correct to determine BPV types before prophylactic (vaccine, etc.) applications.

The Anti-Tumorigenic Role of Cannabinoid Receptor 2 in Non-Melanoma Skin Cancer.

Five million non-melanoma skin cancers occur globally each year, and it is one of the most common malignant cancers. The dysregulation of the endocannabinoid system, particularly cannabinoid receptor 2 (CB2), is implicated in skin cancer development, progression, and metastasis. Comparing wildtype (WT) to systemic CB2 knockout (CB2-/-) mice, we performed a spontaneous cancer study in one-year old mice, and subsequently used the multi-stage chemical carcinogenesis model, wherein cancer is initiated by 7,12-dimethylbenz[a]anthracene (DMBA) and promoted by 12-O-tetradecanoylphorbol-13-acetate (TPA). We found that aging CB2-/- mice have an increased incidence of spontaneous cancerous and precancerous skin lesions compared to their WT counterparts. In the DMBA/TPA model, CB2-/- developed more and larger papillomas, had decreased spontaneous regression of papillomas, and displayed an altered systemic immune profile, including upregulated CD4+ T cells and dendritic cells, compared to WT mice. Immune cell infiltration in the tumor microenvironment was generally low for both genotypes, although a trend of higher myeloid-derived suppressor cells was observed in the CB2-/- mice. CB2 expression in carcinogen-exposed skin was significantly higher compared to naïve skin in WT mice, suggesting a role of CB2 on keratinocytes. Taken together, our data show that endogenous CB2 activation plays an anti-tumorigenic role in non-melanoma skin carcinogenesis, potentially via an immune-mediated response involving the alteration of T cells and myeloid cells coupled with the modulation of keratinocyte activity.

Malignant transformation of a solitary tracheal papilloma in the absence of human papillomavirus infection in a patient with 48, XYYY syndrome: case report.

The diagnosis and management of tracheobronchial papilloma is challenging due to its rarity, and non-specific presenting symptoms. Small percentage undergoes malignant transformation. Herein, we report an unusual case of tracheal papilloma initially misdiagnosed as chronic obstructive pulmonary disease (COPD) in 36-year-old male with triple Y syndrome. It was successfully treated with local debridement and brachytherapy. To the best of our knowledge, this is the first description of brachytherapy for such a condition.

Clinical features and factors affecting prognosis and partial deterioration of ocular papilloma: a retrospective study of 298 cases.

PURPOSE: This study aimed to analyze the clinicopathological features of ocular papilloma, a common benign tumor, and the risk factors related to its recurrence and partial deterioration. METHODS: We collected and analyzed the clinical information of 298 patients (51.68% males) with mean age of 41.54 ± 21.95 years, in the ophthalmology department of the West China Hospital. Clinical and pathological factors that might be related to papilloma recurrence and partial deterioration were studied. RESULTS: The top three papilloma sites were bulbar conjunctiva, eyelid skin and palpebral conjunctiva. Moreover, 3.59% of lesions presented a malignant transformation, and 16.28% of patients had one or more recurrences after an average follow-up of 4.47 years. The multivariate logistic regression model showed that multiple lesions were a risk factor for recurrence (p = 0.022, OR = 3.088, 95% CI: 1.180-8.079), while cryotherapy decreased the recurrence risk (p = 0.044, OR = 0.364, 95% CI: 0.136-0.972). Additionally, elderly patients and lesions on the corneal limbus or cornea had a higher risk of malignant transformation (p = 0.004 and 0.01, OR = 1.086 and 7.827, 95% CI: 1.027-1.150 and 1.629-37.596, respectively). CONCLUSION: Ocular papilloma usually occurs in middle-aged and young patients, with no significant gender differences. Older patients and lesions on the corneal limbus or cornea are risk factors for partial malignant transformation. Finally, multiple lesions were a risk factor for recurrence, and cryotherapy reduced the recurrence rate.

Clinical correlation analysis of HPV infection in adult laryngeal papilloma and adverse events.

OBJECT: To explore the factors affecting dysplasia and carcinogenesis in adult patients with laryngeal papilloma, and the clinical differences between human papillomavirus (HPV)-positive and HPV-negative patients. METHODS: Clinical data of 80 adult patients with laryngeal papilloma and associated adverse events were collected retrospectively. They had undergone surgery in the Department of Otolaryngology Head and Neck, Beijing Tongren Hospital, Capital Medical University between January 2010 and December 2020. HPV infection was detected using RNA in situ hybridization. RESULTS: Regression analysis showed that multiple lesions and high Ki-67 expression were independent factors affecting the occurrence of adverse events. Differences between the HPV-positive and HPV-negative groups were compared. The age and Ki-67 expression in the HPV-negative group were significantly higher than those in the HPV-positive group. In the severe dysplasia to carcinogenesis subgroup, the proportion of HPV-negative patients was significantly higher than that in the mild to moderate dysplasia subgroup. There was a high correlation between positive p16 immunohistochemistry (IHC) and positive HPV. CONCLUSIONS: Multiple lesions and high Ki-67 expression are independent factors that are linked with adverse laryngeal papilloma progression. Elderly HPV-negative patients are at an increased risk of severe dysplasia and carcinogenesis. Positive p16 IHC was very accurate in detecting HPV infection.