scholarly journals Juvenile-Onset Recurrent Respiratory Papillomatosis Aggressiveness: In Situ Study of the Level of Transcription of HPV E6 and E7

Cancers ◽  
2020 ◽  
Vol 12 (10) ◽  
pp. 2836 ◽  
Author(s):  
Charles Lépine ◽  
Thibault Voron ◽  
Dominique Berrebi ◽  
Marion Mandavit ◽  
Marine Nervo ◽  
...  

Juvenile-onset recurrent respiratory papillomatosis (JoRRP) is a condition related to HPV 6 and 11 infection which is characterized by the repeated growth of benign exophytic papilloma in the respiratory tract. Disease progression is unpredictable: some children experience minor symptoms, while others require multiple interventions due to florid growth. The aim of this study was to explore the biomarkers of JoRRP severity on a bicentric cohort of forty-eight children. We performed a CISH on the most recent sample of papilloma with a probe targeting the mRNA of the E6 and E7 genes of HPV 6 and 11 and an immunostaining with p16INK4a antibody. For each patient HPV RNA CISH staining was assessed semi-quantitatively to define two scores: 1+, defined as a low staining extent, and 2+, defined as a high staining extent. This series contained 19 patients with a score of 1+ and 29 with a score of 2+. Patients with a score of 2+ had a median of surgical excision (SE) per year that was twice that of patients with a score of 1+ (respectively 6.1 versus 2.8, p = 0.036). We found similar results with the median number of SE the first year. Regarding p16INK4a, all patients were negative. To conclude, HPV RNA CISH might be a biomarker which is predictive of disease aggressiveness in JoRRP, and might help in patient care management.

2019 ◽  
Vol 98 (1) ◽  
pp. 28-31 ◽  
Author(s):  
Sandipta Mitra ◽  
Arindam Das ◽  
Debasish Ghosh ◽  
Arunabha Sengupta

A prospective observational study was conducted consisting of 21 patients of Juvenile-onset recurrent respiratory papillomatosis, attending the Department of Otorhinolaryngology and Head Neck Surgery at our institution, who underwent surgical excision of the papillomas followed by oral acyclovir postoperatively. The study was aimed to observe the effect of systemic acyclovir on postoperative outcomes in children having recurrent respiratory papillomatosis undergoing primary surgical excision. It was observed that the mean interval between surgeries as well as the number of surgical interventions required was significantly lesser when acyclovir was used as a postoperative adjuvant than when surgery was done alone. Hence, the interval between successive surgeries, or in other words, the time interval between relapse of the disease could be prolonged significantly with the use of postoperative systemic acyclovir. Thus, the disease could be controlled for longer periods and repeated surgeries avoided.


2017 ◽  
Vol 217 (2) ◽  
pp. 208-212 ◽  
Author(s):  
Daniel Novakovic ◽  
Alan T L Cheng ◽  
Yvonne Zurynski ◽  
Robert Booy ◽  
Paul J Walker ◽  
...  

Abstract Background Recurrent respiratory papillomatosis is a rare but morbid disease caused by human papillomavirus (HPV) types 6 and 11. Infection is preventable through HPV vaccination. Following an extensive quadrivalent HPV vaccination program (females 12–26 years in 2007–2009) in Australia, we established a method to monitor incidence and demographics of juvenile-onset recurrent respiratory papillomatosis (JORRP) cases. Methods The Australian Paediatric Surveillance Unit undertakes surveillance of rare pediatric diseases by contacting practitioners monthly. We enrolled pediatric otorhinolaryngologists and offered HPV typing. We report findings for 5 years to end 2016. Results The average annual incidence rate was 0.07 per 100000. The largest number of cases was reported in the first year, with decreasing annual frequency thereafter. Rates declined from 0.16 per 100000 in 2012 to 0.02 per 100000 in 2016 (P = .034). Among the 15 incident cases (60% male), no mothers were vaccinated prepregnancy, 20% had maternal history of genital warts, and 60% were first born; 13/15 were born vaginally. Genotyped cases were HPV-6 (n = 4) or HPV-11 (n = 3). Conclusion To our knowledge, this is the first report internationally documenting decline in JORRP incidence in children following a quadrivalent HPV vaccination program.


Cancers ◽  
2021 ◽  
Vol 13 (11) ◽  
pp. 2556
Author(s):  
Mary C. Bedard ◽  
Alessandro de Alarcon ◽  
Yann-Fuu Kou ◽  
David Lee ◽  
Alexandra Sestito ◽  
...  

Juvenile-onset recurrent respiratory papillomatosis (JoRRP) is the most common benign neoplasm of the larynx in children, presenting with significant variation in clinical course and potential for progression to malignancy. Since JoRRP is driven by human papillomavirus (HPV), we evaluated viral factors in a prospective cohort to identify predictive factors of disease severity. Twenty children with JoRRP undergoing routine debridement of papillomas were recruited and followed for ≥1 year. Demographical features, clinical severity scores, and surgeries over time were tabulated. Biopsies were used to establish a tissue bank and primary cell cultures for HPV6 vs. HPV11 genotyping and evaluation of viral gene expression. We found that patients with HPV11+ disease had an earlier age at disease onset, higher frequency of surgeries, increased number of lifetime surgeries, and were more likely to progress to malignancy. However, the amplitude of viral E6/E7 gene expression did not account for increased disease severity in HPV11+ patients. Determination of HPV strain is not routinely performed in the standard of care for JoRRP patients; we demonstrate the utility and feasibility of HPV genotyping using RNA-ISH for screening of HPV11+ disease as a biomarker for disease severity and progression in JoRRP patients.


2021 ◽  
pp. 1-4
Author(s):  
Corrado Tagliati ◽  
Giuseppe Lanni ◽  
Federico Cerimele ◽  
Antonietta Di Martino ◽  
Valentina Calamita ◽  
...  

We present a case of ductal carcinoma in situ within a fibroadenoma. Breast cancer arising within fibroadenoma incidence ranges from 0.125% to 0.02%, and ductal carcinoma in situ is not the most frequent malignancy that can be found within a fibroadenoma. Dynamic contrast-enhanced magnetic resonance imaging showed an oval mass with circumscribed margins and dark internal septations, suspicious for fibroadenoma. According to European Society of Breast Radiology diffusion-weighted imaging consensus, mean apparent diffusion coefficient value obtained by drawing a small region of interest on the lesion apparent diffusion coefficient map showed a low diffusion level. Therefore, ductal carcinoma in situ within a fibroadenoma was diagnosed at final pathology after surgical excision.


2010 ◽  
Vol 37 (8) ◽  
pp. 1756-1762 ◽  
Author(s):  
VINAYA KUNJIR ◽  
ANURADHA VENUGOPALAN ◽  
ARVIND CHOPRA

Objective.To assess the current International League of Associations for Rheumatology (ILAR) classification criteria (Edmonton, 2001) for juvenile idiopathic arthritis (JIA) in Indian patients.Methods.Out of 441 children, 330 with chronic joint pains were diagnosed with juvenile onset chronic inflammatory arthritis and followed in an observational cohort. Our study was carried out from 1994 to 2006 in a community rheumatology clinic. Emphasis was placed on obtaining data required by the ILAR system. Of the original group, 235 children were eventually classified as having JIA; 108 were examined during the first year of illness.Results.We assigned 224 children (95%) to discrete JIA categories: enthesitis-related arthritis (ERA; 36%), oligoarthritis (OLA-persistent; 17%), polyarthritis rheumatoid factor (RF)-negative (17%), polyarthritis RF-positive (12%), systemic arthritis (8%), OLA-extended (4%), and psoriatic arthritis (1%). The remaining 11 children (5%) were classified with undifferentiated arthritis (mostly an overlap due to seropositive RF and/or HLA-B27). The prevalence of ERA (89% HLA-B27-positive) and seropositive RF was unexpectedly high. Although agreement (κ > 0.79) with the American College of Rheumatology criteria and the European Spondylarthropathy Study Group criteria was good to excellent, the ILAR system was found to be more comprehensive and clinically homogeneous. However, some problems appear unique in our scenario.Conclusion.A wide-spectrum phenotype of JIA is demonstrated by an Indian cohort. Although useful, RF and HLA-B27 in this population proved problematic to the ILAR classification.


F1000Research ◽  
2021 ◽  
Vol 10 ◽  
pp. 913
Author(s):  
Julian Hirt ◽  
Abeelan Rasadurai ◽  
Matthias Briel ◽  
Pascal Düblin ◽  
Perrine Janiaud ◽  
...  

Background: In 2020, the COVID-19 pandemic led to an unprecedented volume of almost 3,000 clinical trials registered worldwide. We aimed to describe the COVID-19 clinical trial research agenda in Germany during the first year of the pandemic. Methods: We identified randomized clinical trials assessing interventions to treat or prevent COVID-19 that were registered in 2020 and recruited or planned to recruit participants in Germany. We requested recruitment information from trial investigators as of April 2021. Results: In 2020, 65 trials were completely (n=27) or partially (n=38) conducted in Germany. Most trials investigated interventions to treat COVID-19 (86.2%; 56/65), in hospitalized patients (67.7%; 44/65), with industry funding (53.8%; 35/65). Few trials were completed (21.5%; 14/65). Overall, 187,179 participants were planned to be recruited (20,696 in Germany), with a median number of 106 German participants per trial (IQR 40 to 345).  From the planned German participants, 13.4%  were recruited (median 15 per trial (IQR 0 to 44). Conclusions: The overall German contribution to the worldwide COVID-19 clinical trial research agenda was modest. Few trials delivered urgently needed evidence. Most trials did not meet recruitment goals. Evaluation and international comparison of the challenges for conducting clinical trials in Germany is needed.


2019 ◽  
Author(s):  
Souhir Khemiri ◽  
Jihene Feki ◽  
Afef Khanfir ◽  
Mohamed Abdelmoula ◽  
Mounir Frikha

Abstract- Melanotic neuroectodermal tumor of infancy (MNTI) is a rare and distinctive neoplasm of early infancy with rapid expansile growth and a high rate of recurrences. Most commonly the lesion affects the maxilla of infants during the first year of life, but it may also occur in the mandible, skull, brain, epididymis, and other rare locations. Common treatment methods’ include surgical excision and resection of the tumor. The aim of this article was to show the diagnosis and treatment of a 7-month-old patient with melanotic neuroectodermal tumor occurred in the anterior mandible and to demonstrate the effectiveness of the neoadjuvant chemotherapy.


2021 ◽  
Vol 47 ◽  
pp. e627
Author(s):  
Kristhian Franchesco Paul PINAZO Beltran ◽  
Jesús Miguel Angel BERRÚ Beltran ◽  
Edwin Fredy BOCARDO Delgado

The prawn (Cryphiops caementarius) is the only resource in the Peruvian coastal rivers that supports a commercial fishery. Bioeconomic-fishing aspects are reported based on data (fishing, costs and income derived from fishing) acquired in situ monthly in four altitudinal strata (every 200 meters above sea level - masl) of the Majes-Camaná river during 2019. The catch per unit of effort (CPUE) was expressed in kg h-1, monthly income was estimated based on the average catch values, number of tasks and price of the resource. To evaluate the profitability of the activity of an average fisherman (by stratum), a cash flow based on income and expenses was executed under situations with a constant future, using economic profitability indicators such as the Net Present Value (NPV), Internal Rate of Return (IRR), Benefit-Cost ratio (B/C) and Discounted Payback Period (DPB). There was a predominance of the diving method, the catches (kg) and the fishing yield increased throughout the fishing period (April-December) with a maximum value of 4.8 kg h-1 (December - high strata). Investment costs per fisherman were low, roughly 1,000 soles. The sale price increases with altitude, decreasing over the course of the year depending on the availability of the resource; the monthly income per fisherman varied from 964 soles (April - low stratum) to 6,760 soles (December - high stratum). The economic simulation model showed that the income exceeded the costs of the fishing activity from the first year of activity in all the altitudinal strata, the economic profitability indicators showed high profitability for the activity.


2019 ◽  
Author(s):  
Edward H. Bair ◽  
Karl Rittger ◽  
Jawairia A. Ahmad ◽  
Doug Chabot

Abstract. Ice and snowmelt feed the Indus and Amu Darya rivers, yet there are limited in situ measurements of these resources. Previous work in the region has shown promise using snow water equivalent (SWE) reconstruction, which requires no in situ measurements, but validation has been a problem until recently when we were provided with daily manual snow depth measurements from Afghanistan, Tajikistan, and Pakistan by the Aga Khan Agency for Habitat (AKAH). For each station, accumulated precipitation and SWE were derived from snow depth using the SNOWPACK model. High-resolution (500 m) reconstructed SWE estimates from the ParBal model were then compared to the modeled SWE at the stations. The Alpine3D model was then used to create spatial estimates at 25 km to compare with estimates from other snow models. Additionally, the coupled SNOWPACK and Alpine3D system has the advantage of simulating snow profiles, which provide stability information. Following previous work, the median number of critical layers and percentage of facets across all of the pixels containing the AKAH stations was computed. For SWE at the point scale, the reconstructed estimates showed a bias of −42 mm (−19 %) at the peak. For the coarser spatial SWE estimates, the various models showed a wide range, with reconstruction being on the lower end. For stratigraphy, a heavily faceted snowpack is observed in both years, but 2018, a dry year, according to most of the models, showed more critical layers that persisted for a longer period.


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