Successful Antiviral Treatment for Chronic Adenoiditis Caused by Herpes Virus in a Pediatric Patient: A Case Report.

BACKGROUND The adenoids are lymphatic tissue located in the nasopharynx and play a role in upper-airway immunity. Inflammation of the adenoids is called adenoiditis, which can cause a variety of symptoms. This is a common condition and is due to acute viral or bacterial infection. Most patients experience mild symptoms of upper-respiratory tract infection with a self-limiting course. CASE REPORT A 5-year-old female patient was brought into the clinic by her parents with concerns regarding hearing and sleep. Clinical assessment was consistent with persistent otitis media with effusion and sleep-disordered breathing. She was scheduled for surgery, including nasendoscopy, adenoidectomy, and bilateral grommet insertion. During surgery, direct visualization of the postnasal space showed complete obstruction by hypertrophic, inflamed adenoids covered in a thick, white film. A biopsy was taken, which detected herpes virus cytopathic effect. A diagnostic workup excluded a neoplastic process and other bacterial or fungal infections. A trial of oral antiviral medication was successful and follow-up nasendoscopy showed resolution of adenoid hypertrophy. CONCLUSIONS Direct visualization of the postnasal space, with a transoral mirror or 120-degree endoscope, prior to adenoidectomy can aid diagnosis. Adenoiditis may be caused by a wide range of organisms, including herpes virus. Active mucopurulent discharge should raise concern for infection by bacteria, fungi, or virus. Previous research on viral infection of the adenoids have been in asymptomatic patients with presumed latent infection and undergoing elective adenoidectomy. To our knowledge, this is the first paper to report on successful treatment with antiviral medication alone.

A Comparative Evaluation of TRPS1 and GATA3 in adenoid cystic, secretory, and acinic cell carcinomas of the breast and salivary gland.

GATA3 is the most used marker to determine tumors' breast origin, but its diagnostic value in triple-negative breast cancer (TNBC) is limited. The newly identified TRPS1 is highly sensitive and specific for breast carcinoma, especially TNBC. Here, we compared the utility of TRPS1 and GATA3 expression in a subset of salivary gland-type breast tumors (including adenoid cystic, acinic cell, and secretory carcinomas [AdCC, ACC, and SC, respectively]), and we compared TRPS1 and GATA3 expression of such tumors with head and neck (H&N) and AdCC of upper respiratory tumors. TRPS1 was strongly expressed in basaloid TNBC and AdCCs with solid components, including 100 % of mixed and solid breast AdCCs. However, TRPS1 was positive in only 50 % cribriform AdCCs. Expression patterns of TRPS1 in H&N and upper respiratory AdCC were similar. TRPS1 was positive in 30 % of H&N cribriform AdCCs but was strongly expressed in mixed AdCC (67 %) and solid AdCC (100 %). In the upper respiratory AdCCs, TRPS1 was positive in 58.4 % of cribriform AdCCs and positive in 100 % of AdCCs with solid components. On the contrary, GATA3 was negative in predominant AdCCs of the breast, H&N, and upper respiratory tract. These data show that GATA3 and TRPS1 expression varies AdCCs. In addition, TRPS1 and GATA3 expression patterns were similar SC and ACC of breast and H&N. Both markers were positive in SC and negative in ACC. Therefore, TRPS1 and GATA3 cannot be used to differentiate salivary gland-type carcinomas of breast origin from those of upper respiratory or H&N origin.

Cervical Adenoid Basal Carcinoma With High-grade Squamous Component: True Mixed Carcinoma or Colonization of Adenoid Basal Carcinoma by High-grade Squamous Intraepithelial Lesion?

Adenoid basal carcinoma (ABC) is a rare clinically indolent human papillomavirus-associated cervical neoplasm with uniformly bland morphology which in pure form does not metastasize. Many cases co-exist with a human papillomavirus-associated high-grade squamous intraepithelial lesion (HSIL) or squamous cell carcinoma (SCC). The ABC and high-grade squamous components may be clearly separate, albeit intermingled, and when the high-grade squamous component is invasive, the tumor is designated a mixed carcinoma, with clinical behavior determined by the non-ABC component. In other cases, discrete nests of high-grade atypical squamous cells are intimately admixed and incorporated within the ABC. These are more difficult to classify but are also usually reported as mixed carcinomas. Herein, we report a series of 9 cases of ABC in patients aged 33 to 89 years (mean age: 63 y) with a high-grade squamous component. In 7 cases, the high-grade squamous cells partly replaced and expanded the nests of ABC, sometimes with a residual cuff of ABC cells, while in the other 2 cases the ABC and SCC were clearly separate. We propose that the aforementioned 7 cases represent colonization of ABC by HSIL rather than mixed carcinomas; as far as we are aware, this concept has not been proposed before. In all cases which we feel represent colonization of ABC by HSIL, the tumors were confined to the cervix (stages IA1 [3 tumors], IA2 [2 tumors], IB1 [2 tumors]) and follow-up was unremarkable with no evidence of metastasis. One case with separate components of ABC and SCC was stage IVA at diagnosis and the patient died of disease. The other was stage IB1 at diagnosis and the patient died of unrelated causes at 13 months. We discuss the clinical implications of distinguishing true mixed carcinomas from colonization of ABC by HSIL and provide an approach to diagnosis. We also report a unique case of colonization of so-called cervical ectopic prostatic tissue by HSIL.

Nasopharyngeal cyst in an adolescent boy: differential diagnosis - embryological and anatomical considerations.

In the current article, we present a case of an adolescent boy with a nasopharyngeal cyst that induced nasal and Eustachian tube obstruction. Nasopharyngeal cysts can be found incidentally during imaging examinations such as MRI; however, a symptomatic nasopharyngeal cyst is a rare finding in the paediatric population. The cyst was treated successfully by marsupialisation, and the histological diagnosis revealed an adenoidal retention cyst. The differential diagnosis of a nasopharyngeal cyst is always challenging since developmental cysts such as Rathke's pouch cysts, Torwaldt's and branchial cleft cysts may be encountered at the nasopharynx. The current article also intends to present the diagnostic and therapeutic approach to a nasopharyngeal cyst, emphasising anatomical and embryological considerations that address its differential diagnosis.

Olfactory disorders in childhood: A comparative study of olfaction in children with adenoid hyperplasia versus a control group and the postoperative effects of adenoidectomy with respect to olfactory ability.

PURPOSE: Hyposmia in childhood is poorly characterized. The "U-Sniff Test", validated for children with anosmia, can be used to objectify olfactory impairment but has not been used to distinguish between hyposmia and normosmia. Therefore, we investigated children with enlarged adenoids with respect to hyposmia, its correlation with adenoid size, and the sensitivity of questionnaires to predict olfactory impairment. METHODS: In a prospective comparison, olfaction was assessed by "U-Sniff Test" (score 0-12; <8 hyposmia) in 41 children (5-18 years) with adenoid hyperplasia and compared with 196 children without any respiratory affection (control) after exclusion of previous SARS-Cov2-infection from December 2020 to December 2021. ENT-related complaints were collected using a self-designed questionnaire. We were able to include 13 children in a follow-up examination to compare preoperative performance in the "U-Sniff Test" with postoperative outcome after adenoidectomy. STATISTICS: chi-square-test (p < 0.05), odds-ratio, Spearman's rho, ROC-, cluster analysis. RESULTS: Severe hyposmia was present in 36.6% of children with adenoid-hyperplasia compared to 3.1% of the control-group. Adenoid-children scored significantly more often between 8 and 10 points (58.5%) than the control (31.6%; p < 0.01). Adenoid size and olfactory performance correlate significantly (r: 0.83; CI -0.89 … -0.72). Hyposmia in the adenoid group is characterized predominately by loss of the odors banana, butter and rose. None of children with hyposmia or parents reported impaired olfactory performance. Postoperatively, olfactory function improved significantly in 85% of cases (p 0.01, SD ± 1.71, Δ3.54points). CONCLUSION: Questionnaires are insufficient to detect hyposmia in this cohort. In contrast, the "U-Sniff Test" detects even reduced olfactory performance without reaching the cut-off value, which represents the majority of test results in the adenoid group. Therefore, we recommend the classification of moderate hyposmia (8-10 points) to be included for our study population.

Adenoid 'ameloblastoma': Clinicopathological description of 4 additional BRAF-negative cases.

OBJECTIVE: Adenoid ameloblastoma (AA) is an epithelial odontogenic tumor that was recognized as a separate entity in the last odontogenic classification of WHO in 2022. The etiology is unknown, and the pathogenesis remains controversial. The objective of this study is to contribute the clinicopathological features of 4 additional BRAF-negative cases to the existing literature, aiming to enhance the molecular understanding of this unique tumor in the forthcoming classification. MATERIALS AND METHODS: This study consists of a case series of four patients diagnosed with AA. The patients' demographic and clinical information were collected from the universities' medical achieves. Histopathologically, all cases were reexamined according to the latest update of the WHO odontogenic tumor classification. In addition to H&E and immunohistochemical stains, cytogenetics was also evaluated. RESULTS: Well-defined unilocular radiolucent lesions were observed in all cases. Ameloblastoma-like components exhibited reserved nuclear polarity, suprabasal stellate reticulum-like epithelium, duct-like structure, whorls/morules, and cribriform architecture were common features. Variable immunoreactivity to CK7, CK19, CK14, p63, and p40 were determined, and proliferative activity was greater than 15%. The BRAF molecular study revealed no mutations. CONCLUSIONS: When diagnosing AA, the essential histopathological characteristics must be rigorously applied, and a significant portion of the lesion should contain these features. Additionally, despite limited molecular data, since the BRAF mutation commonly observed in ameloblastomas is not present in the majority of AA cases, we propose changing the term "ameloblastoma" to "ameloblastic" and referring to it as "adenoid ameloblastic tumor" in the forthcoming classification.

A Systematic Review of Adenoid Ameloblastoma: A Newly Recognized Entity.

BACKGROUND: Recently, a new odontogenic tumor has been described, the so-called adenoid ameloblastoma (AdAM). The aim of this review was to determine the clinical and imaging features of AdAM and to describe its main histopathological findings. METHODS: The systematic review included published cases with a diagnosis of AdAM in the gnathic bones, which had sufficient clinical, imaging, and histopathological data to confirm its diagnosis. The following histopathological diagnostic criteria were adopted: presence of ameloblastoma-like components, duct-like structures, spiral cellular condensations, and a cribriform architecture. RESULTS: Fifteen articles, corresponding to 30 cases of AdAM, were selected. Most cases affected men (63.3%), with a slight preference for the mandible (16:14) and the posterior region of gnathic bones was the most commonly affected site. The mean age at diagnosis was 40.8 years. Clinically, the lesions usually presented as a swelling (53.3%) and, radiographically, as a well-defined radiolucency (33.4%). Surgical resection (40%) was the most frequently adopted treatment and recurrence occurred in 30% of cases. Microscopic examination showed cribriform areas in most AdAM cases (93.3%); duct-like structures and spiral cellular condensations were seen in 100% of the cases. CONCLUSION: The small number of reported cases, the existence of erroneous diagnoses, and the adoption of initial conservative management make it difficult to determine whether AdAM has a higher risk of recurrence or more aggressive biological behavior than conventional ameloblastomas.

Oropharyngeal microbiome in children with adenotonsillar hypertrophy and after adenotonsillectomy.

BACKGROUND: The microbiome of tonsils and adenoid in adenotonsillar hypertrophy (ATH) has been profiled. Adenotonsillectomy (AT) is widely used for ATH in children. The variation of the oropharyngeal microecology in children with ATH or after AT have never been studied. OBJECTIVES: Here we aimed to evaluate the change in oropharyngeal microbiome in ATH Children after AT. MATERIALS AND METHODS: In this cross-sectional study, throat swabs for microbiome analysis were collected from ATH, AT and control groups. Using 16s rDNA sequencing, this study investigated the characteristics of oropharyngeal microbiome. RESULTS: The α-diversity showed a statistical difference in richness and the ß-diversity was significantly different among the three groups. The relative abundance of Haemophilus (member of Proteobacteria) increased but that of Actinomyces (member of Actinobacteriota) decreased in the ATH group compared to those in the AT and control groups, but their abundances showed no statistical difference between the AT and control groups. CONCLUSIONS: The oropharyngeal microbial diversity and composition are disrupted in children with ATH and can be restored after AT. This microbiome analysis provides a new understanding about the pathogenesis of ATH in children.SummaryIn this study, we aimed to evaluate the change in oropharyngeal microbiome in ATH Children after AT. The oropharyngeal microbial diversity and composition are disrupted in children with ATH and can be restored after AT.

Adenoid hypertrophy-​diagnosis and treatment: the new S2k guideline.

Hyperplasia of the pharyngeal tonsils is to be considered pathologic when nasopharyngeal symptoms of mechanical obstruction and/or chronic inflammation occur. Chronic Eustachian tube dysfunction can result in various middle ear diseases such as conductive hearing loss, cholesteatoma, and recurrent acute otitis media. During examination, attention should be paid to the presence of adenoid facies (long face syndrome), with a permanently open mouth and visible tip of the tongue. In the case of severe symptoms and/or failure of conservative treatment, adenoidectomy is usually performed on an outpatient basis. Conventional curettage remains the established standard treatment in Germany. Histologic evaluation is indicated for clinical evidence of mucopolysaccharidoses. Due to the risk of hemorrhage, the preoperative bleeding questionnaire, which is obligatory before every pediatric surgery, is referred to. Recurrence of adenoids is possible despite correct adenoidectomy. Before discharge home, otorhinolaryngologic inspection of the nasopharynx for secondary bleeding should be performed and anesthesiologic clearance obtained.

Role of Mesenchymal Stem Cells on Tonsillar Hypertrophy: An Unexplored Enigma.

Tonsillar or adenoid hypertrophy is a common childhood finding which can cause significant health problems like respiratory infections and sleep apnea. Though normal growth of children is also attributed to such enlargement, infection, environmental pollutants, allergens, and gastroesophageal reflux are proposed triggering factors for tonsillar hypertrophy. While tonsilar enlargement in adults is more associated with malignancy and chronic infections like the human immunodeficiency virus, the immunology of childhood adenotonsillar hypertrophy is less understood. We postulate that upon stimulation, mesenchymal stem cells are found to reduce the secretion of interferon-gamma but increase the secretion of interleukin-4 from activated T cells. Both of these factors inhibit apoptosis in the tonsillar tissue leading to its hypertrophy. Under the umbrella of evidence, it implicates the role of mesenchymal stem cells in tonsillar hypertrophy. However, further longitudinal large studies are needed to validate the proposition. Keywords: interleukin-4; mesenchymal stem cell; tonsillar hypertrophy.

[Adenoids-diagnosis and treatment: the new German S2k guideline]. / Adenoide Vegetationen ­ Diagnostik und Therapie ­ die neue S2k-Leitlinie.

Hyperplasia of the pharyngeal tonsils is to be considered pathologic when nasopharyngeal symptoms of mechanical obstruction and/or chronic inflammation occur. Chronic Eustachian tube dysfunction can result in various middle ear diseases such as conductive hearing loss, cholesteatoma, and recurrent acute otitis media. During examination, attention should be paid to the presence of adenoid facies (long face syndrome), with a permanently open mouth and visible tip of the tongue. In the case of severe symptoms and/or failure of conservative treatment, adenoidectomy is usually performed on an outpatient basis. Conventional curettage remains the established standard treatment in Germany. Histologic evaluation is indicated for clinical evidence of mucopolysaccharidoses. Due to the risk of hemorrhage, the preoperative bleeding questionnaire, which is obligatory before every pediatric surgery, is referred to. Recurrence of adenoids is possible despite correct adenoidectomy. Before discharge home, otorhinolaryngologic inspection of the nasopharynx for secondary bleeding should be performed and anesthesiologic clearance obtained.

Adenoid hypertrophy in children: a narrative review of pathogenesis and clinical relevance.

Adenoids (nasopharyngeal tonsils), being part of Waldeyer's ring, are masses of lymphoid tissues located at the junction of the roof and the posterior wall of the nasopharynx. Adenoids play an important role in the development of the immune system and serve as a defence against infections, being the first organs that come into contact with respiratory and digestive antigens. The causes of adenoid hypertrophy are not fully known. They are most likely associated with aberrant immune reactions, infections, environmental exposures and hormonal or genetic factors. The aim of this review is to summarise the current knowledge of adenoid hypertrophy in children and associated diseases. Adenoid hypertrophy has many clinical manifestations that are frequent in the paediatric population and is accompanied by various comorbidities.

Adenoid Ameloblastoma with BRAF p.V600E Mutation Revealing Ameloblastomatous Origin: A First Case Report.

BACKGROUND: Adenoid ameloblastoma (AdAM) is a frequently recurrent tumor that shows hybrid histological features of both ameloblastoma and adenomatoid odontogenic tumor (AOT). AdAM is expected to be classified as a new subtype of ameloblastoma in the next revision of the World Health Organization (WHO) odontogenic tumor classification. However, whether AdAM is a histologic variant of ameloblastoma or AOT remains unclear. To establish a new category, genetic evidence indicating the tumor category is necessary. METHODS: We present a case of a 23-year-old Japanese woman with AdAM who underwent genetic/DNA analysis for ameloblastoma-related mutation using immunohistochemical staining, Sanger sequencing, and next-generation sequencing (NGS) analyses with reliable clinicopathological evidence. RESULTS: Immunohistochemical expression of BRAF p.V600E was diffusely positive for both ameloblastoma- and AOT-like components. Sanger sequencing and NGS analyses showed missense mutations in BRAF p.V600E (c.1799T > A), a gene that is commonly altered in ameloblastomas but not in KRAS, another gene associated with AOT. CONCLUSION: This case report is the first to provide genetic evidence on the ameloblastomatous origin of AdAM with a BRAF p.V600E mutation. A larger series of AdAM groups' molecular testing is needed to aptly classify them and prognosticate the best treatment.

A case of metastatic adenoid cystic (basal cell) carcinoma of the prostate: Systemic therapy for a rare disease.

BACKGROUND: Metastatic adenoid cystic (basal cell) carcinoma of the prostate is an exceedingly rare disease entity. As a result, no current consensus exists for optimal systemic therapy. METHODS: We present a patient with metastatic adenoid cystic (basal cell) carcinoma of the prostate who subsequently received systemic treatment, including chemotherapy and immunotherapy. We comprehensively reviewed all published data on therapy outcomes in advanced disease. RESULTS: Our patient benefited from combination chemotherapy (carboplatin and paclitaxel), with objective radiographic response and reduction in cancer-related pain. However, chemotherapy was stopped due to cumulative neurotoxicity, and subsequent immunotherapy with atezolizumab did not produce any response. Our literature review revealed inconsistent outcomes with various treatments but showed most promise with chemotherapy. Targeted therapy and immunotherapy seem to benefit specific cases, and androgen deprivation therapy had minimal evidence of benefit. CONCLUSION: Based on the findings of our case report and literature review, we suggest platinum-based chemotherapy doublets as first-line treatment for metastatic cases of adenoid cystic (basal cell) carcinoma of the prostate, reserving targeted therapy or immunotherapy for select cases based upon molecular profiles.

Adenoid Ameloblastoma Shares Clinical, Histologic, and Molecular Features With Dentinogenic Ghost Cell Tumor: The Histologic Spectrum of WNT Pathway-Altered Benign Odontogenic Tumors.

An epithelial odontogenic tumor called adenoid ameloblastoma (AA) has recently been included in the new WHO classification. However, AA has considerable overlapping features with a preexisting entity, dentinogenic ghost cell tumor (DGCT). This study compared the clinical, histologic, and molecular characteristics of AA and DGCT. Eight cases of odontogenic tumors initially diagnosed as AA or DGCT were included in this study. Quantitative histologic analysis, ß-catenin immunohistochemistry, and molecular profiling using next generation sequencing were performed. Additionally, accumulated clinical data of AA and DGCT were statistically analyzed. Nuclear ß-catenin accumulation was detected in all cases in common, although the tumors studied histologically consisted of varying combinations of the AA-like phenotype, ghost cells, and dentinoid. However, CTNNB1 hotspot mutations were not found in any case. Instead, loss-of-function mutations in tumor suppressor genes involved in the WNT pathway, including the APC, SMURF1, and NEDD4L genes, were found regardless of histologic type. In addition, KRT13 mutations were detected in 2 cases with a high proportion of ghost cells. Finally, a literature analysis revealed clinical similarities between the previously reported cases of AA and DGCT. These findings suggest that from a clinical and molecular point of view, AA and DGCT represent a histologic spectrum of WNT pathway-altered benign odontogenic tumors rather than 2 distinct tumors. Moreover, previously unidentified keratin mutations may be associated with ghost cell formation found in specific types of odontogenic lesions.

Unilateral tonsil enlargement in children and adults: is routine histology tonsillectomy warranted? A multi-centre series of 323 patients.

BACKGROUND: This study aimed to establish whether histology tonsillectomy is justified for unilateral tonsil enlargement. METHODS: A retrospective review was conducted of histology tonsillectomies in three health organisations over five years, with strict exclusion criteria, focusing on benign-appearing unilateral tonsil enlargement. RESULTS: Ninety paediatric and 233 adult cases were included. No paediatric cases and five adult cases of malignancy were detected. All malignant cases presented with other symptoms. Using binary logistic regression, a history of rapid unilateral tonsil enlargement was the only factor found to be significantly associated with malignant outcome. Thirty-three per cent of subjectively larger tonsils were smaller on post-operative histological measurement. Of the cases, 12.1 per cent re-presented with post-tonsillectomy bleeding. CONCLUSION: The authors recommend avoiding histology tonsillectomy for unilateral tonsil enlargement unless 'red flag' signs of malignancy are present, with particular attention to rapid unilateral tonsil enlargement. This study demonstrated discrepancy between clinical examination findings and true tonsil asymmetry; there may be a role for cross-sectional imaging prior to histology tonsillectomy in high-risk patients.

Adenoid Ameloblastoma Versus Dentinogenic Ghost Cell Tumor.

BACKGROUND: A recent systematic review published in Head and Neck Pathology found that 3.8% of dentinogenic ghost cell tumors harbor duct-like/ cribriform architecture. Herein we discuss this finding regarding the differential diagnosis of this tumor with adenoid ameloblastoma. METHODS: A critical review of some microscopic findings reported in a recent paper published in the Head and Neck Pathology Journal was done. RESULTS: Although there are overlapping microscopic features with dentinogenic ghost cell tumor, adenoid ameloblastoma is distinguished by the combination of duct-like structures and whorls/morules. In our opinion, at least some cases previously diagnosed as dentinogenic ghost cell tumors may now be more accurately classified as adenoid ameloblastoma. CONCLUSION: We conclude that a reassessment of dentinogenic ghost cell tumor cases using the diagnostic criteria proposed by the new WHO classification of Head and Neck Tumors (2022) is warranted.