scholarly journals Recurrent Respiratory Papillomatosis (RRP)—Juvenile Onset

2008 ◽  
Vol 2 ◽  
pp. CMO.S698 ◽  
Author(s):  
Sara W. Dyrstad ◽  
Krishna A. Rao

In this article, we describe the treatment of long standing juvenile-onset recurrent respiratory papillomatosis (JORRP) with eventual transformation to carcinoma in a patient who lived to the age of 73. Treatment modalities consisted of bronchoscopy and local excision initially. Later, YAG and CO2 laser debulking were used. Radiotherapy, chemotherapy with carboplatin (300 mg/m2) and 5-FU (600 mg/m2), oral methotrexate (5–7.5 mg/week), pegylated Interferon, indole-3-carbamide, and intralesional cidofovir were also utilized in the treatment of this patient. Except for methotrexate, each of the treatment regimens used in this patient, initially decreased growth of the papillomas and improved symptoms experienced by the patient. Interestingly, we found that this patient's long standing JORRP initially responded to a chemotherapy regimen of 4 cycles of carboplatin (300 mg/m2) and 5-FU (600 mg/m2) as well. Ultimately, the disease became resistant to all forms of treatment and progressed. The patient eventually succumbed to the disease after an approximate 77 year course.

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.


QJM ◽  
2021 ◽  
Vol 114 (Supplement_1) ◽  
Author(s):  
Mohamed Abdullah Eshafi ◽  
Nehal Mohamed Zuelfakkar ◽  
Ahmed Abd Elfattah Afify

Abstract Vitiligo is a disease that causes the loss of skin color in patches due to loss of melanin pigmentation of specific areas of the skin. Although several hypotheses have been proposed, the leading theory is still the auto-immune etiology linked to specific genetic mutations. Vitiligo can also be associated with several autoimmune diseases. There is no curative treatment for vitiligo but, several treatment modalities are considered. Topical therapies like steroids and Calcineurin inhibitors are of popular use in clinical settings also, steroids can be administered systemically in vitiligo patients. Physical therapies as fractionated CO2 (Fr: CO2) laser and Narrowband-UV (NBUV) phototherapy represent a gold standard in treatment in clinical practice. Moreover, intralesional therapies are emerging, one of which is autologous platelet-rich plasma injection. Aim of the study This study aimed to evaluate and compare the efficacy and safety of Fr: CO2 laser, PRP, combined Fr: CO2 laser and PRP, combined Fr: CO2 laser and NB-UVB, combined Fr: CO2 laser, PRP and NB-UVB in the treatment of vitiligo as well as reporting the side effects. Patients and methods This study included 20 vitiligo patients with at least 6 patches of stable vitiligo (120 patches), the patches were divided into six groups according to the treatment modality. Assessment of treatment response was done through patient satisfaction score and Vitiligo analysis by computer-assisted grid (VACAG). Results Regarding surface area reduction in included patients, fractional CO2 laser achieved the best results followed by triple combination therapy (CO2 with PRP and NB-UVB), the least response was with CO2 with PRP treatment. Patient satisfaction in the current study had a different outcome, PRP treated patients exhibited the highest satisfaction scores while triple combination treated group showed the least satisfaction scores.


Cancers ◽  
2020 ◽  
Vol 12 (11) ◽  
pp. 3414
Author(s):  
Hye Won Lee ◽  
Dai Hoon Han ◽  
Hye Jung Shin ◽  
Jae Seung Lee ◽  
Seung Up Kim ◽  
...  

By pegylated interferon (PegIFN)-free direct-acting antivirals (DAAs) against hepatitis C virus (HCV) infection, a sustained virological response (SVR) rate >95% can be attained with a satisfactory tolerability and shorter treatment duration. However, it remains controversial whether there is any difference in prognosis depending on regimens—PegIFN or DAAs. We compared the probabilities of hepatocellular carcinoma (HCC) development between patients achieving an SVR by PegIFN/ribavirin (PegIFN group, n = 603) and DAAs (DAAs group, n = 479). The DAAs group was significantly older and had a higher proportion of cirrhosis than the PegIFN group. Before adjustment, the DAAs group had a higher HCC incidence than the PegIFN group (p < 0.001). However, by multivariate analyses, the DAAs (vs. PegIFN) group was not associated with HCC risk (adjusted hazard ratio 0.968, 95% confidence interval 0.380–2.468; p = 0.946). Old age, male, higher body mass index, cirrhosis, and lower platelet count were associated with increased HCC risk (all p < 0.05). After propensity score matching (PSM), a similar HCC risk between the two groups was observed (p = 0.372). We also compared HCC incidences according to sofosbuvir (SOF)-based and SOF-free DAAs, showing a similar risk in both groups before adjustment (p = 0.478) and after PSM (p = 0.855). In conclusion, post-SVR HCC risks were comparable according to treatment regimens; PegIFN- vs. DAA-based regimens and SOF-based vs. SOF-free DAA regimens. Further studies with a longer follow-up period are required.


2019 ◽  
Vol 53 (2) ◽  
Author(s):  
Shahara Abalos-Babaran ◽  
Jay-V James G. Barit ◽  
Francisca DC. Roa

Background and Objectives. Potassium hydroxide (KOH) is effective and safe as treatment of viral dermatoses. No systematic review has been done reporting its efficacy as a treatment for anogenital warts. Methods. A systematic literature search for controlled clinical trials using KOH, any drug or ablative procedure measuring the clearance rate was conducted. Analysis was done using RevMan v5.3 software. Results. Four low quality trials, composed of 197 patients were included but only two qualified for meta-analysis. Two studies compared KOH to cryotherapy while the two other trials compared KOH to intralesional 5-fluorouracil (FU) + salicylic acid (SA) and carbon dioxide (CO2) laser vaporization. The KOH group showed a higher clearance rate compared to cryotherapy (RR= 1.40, P> 0.05, I2=39 %) and no recurrence was noted (RR= 0.17, P> 0.05, I2=0) but the difference is not statistically significant. Isik et al., 2014 and Asadi et al., demonstrated that there was no significant difference among groups receiving KOH, 5-FU+SA and CO2 laser vaporization in the mean lesion count and size at follow up visits. (P > 0.05). Conclusions. Potassium hydroxide has comparable efficacy to the present treatment modalities but well-structured RCTs are needed to further support its use.


2021 ◽  
Author(s):  
Martina Bertinazzi ◽  
Tarik Gheit ◽  
Jerry Polesel ◽  
Sandrine McKay-Chopin ◽  
Cesare Cutrone ◽  
...  

Abstract Purpose The aim of our study is to evaluate the prevalence of different HPV genera – alpha, beta and gamma – in Juvenile onset Recurrent Respiratory Papillomatosis (JoRRP) and examine the association of type and genus-specific viral features with the clinical outcome of disease. Methods This retrospective observational study includes consecutive patients with JoRRP who were treated in a referral centre between October 2000 and October 2020. All patients underwent cold excision and laser vaporization of papillomatous lesions. Samples were analysed for the presence of 120 viral genotypes (22 alpha-HPV, 46 beta-HPV, 52 gamma-HPV) using a highly sensitive multiplex genotyping assay. Results Twenty patients with JoRRP, aged 0.3–11 years old, were included, with a median follow-up of 13.5 years. All samples were HPV DNA positive: 20 (100%) for alpha-HPV DNA; 7 (35%) for beta – HPV DNA; 0 for gamma-HPV DNA. Three groups were defined according to the number of infections: seven cases (35%) with HPV mono-infection; ten cases (50%) with HPV double-infection; three cases (15%) with ≥ 3 HPV infections. At diagnosis, patients with ≥ 3 HPV infections reported higher median Derkay’s score than those with mono-infection (21 vs 14, P = 0.018). Number of HPV infections was also associated with clinical outcomes, with an average of 0.5 surgical procedures/year in patients with mono-infection, 1.2 for double-infection, 2.6 for ≥ 3 infections (P = 0.006). Conclusion Despite the small sample size, these preliminary data support an association between the number of different alpha and beta HPV co-infections and the clinical severity of the disease.


2018 ◽  
Vol 22 (6) ◽  
pp. 591-601 ◽  
Author(s):  
Linghong Linda Zhou ◽  
Jorge R. Georgakopoulos ◽  
Arvin Ighani ◽  
Jensen Yeung

Generalized pustular psoriasis (GPP) is a rare but serious and difficult to treat cutaneous disease, with high morbidity and mortality rates. Despite the numerous treatment regimens available, the overall quality of evidence-based research is limited with a lack of an algorithmic approach available. In this review, we aim to evaluate the current level of evidence regarding the efficacy and safety/tolerability of systemic monotherapies available in the treatment of GPP. A comprehensive MEDLINE, EMBASE, and PubMed search of clinical studies examining systemic monotherapy treatment options for GPP was conducted. In total, 31 studies met eligibility criteria. Described treatment modalities included retinoids, cyclosporine, biologics, and dapsone. Despite the lack of high-quality evidence or a well-accepted treatment algorithm for GPP, systemic retinoids, cyclosporine, biologics, and dapsone are all possible first-line agents, with retinoids being one of the best-supported treatment options and biologics as an emerging therapeutic field with great potential requiring additional data. However, the final choice of treatment should be considered within the unique context of each patient.


2021 ◽  
pp. 014556132110498
Author(s):  
Xiaoli Qu ◽  
Yang Xiao ◽  
Lijing Ma ◽  
Jun Wang

Objectives The lesion distribution of juvenile-onset recurrent respiratory papillomatosis (JORRP) during first-time surgery has been rarely reported. The purpose of this study was to describe the anatomical distribution of papilloma across 25 Derkay sites during initial surgery and to assess the impact of the lesion distribution on disease severity. Methods Surgical videos and medical records of 106 patients with JORRP (27 aggressive and 79 nonaggressive cases) were retrospectively reviewed. Lesion locations were recorded using Derkay anatomical sites. Logistic regression was used to analyze the effect of the lesion distribution on disease severity. Results Among the 106 patients, the true vocal cords (90.6% left, 84.0% right) were the most frequently involved site, followed by the false vocal cords (39.6% left, 35.8% right) and the anterior commissure (26.4%). Two patients (1.9%) had tracheal involvement. Patients with false vocal cord involvement (odds ratio [OR] = 3.425, 95% confidence interval [CI] [1.285, 9.132], P = .014) and a younger age at diagnosis (OR = .698, 95% CI [.539, .905], P = .007) were more likely to require more than 4 procedures in the year following first-time surgery. Conclusions Lesions were most common on the true vocal cords. False vocal cord involvement and a younger age at diagnosis were risk factors for disease severity.


2019 ◽  
Vol 34 (7) ◽  
pp. 1433-1440 ◽  
Author(s):  
Joanna Jackowska ◽  
Waldemar Wojnowski ◽  
Anna Hashimoto ◽  
Bogna Małaczyńska ◽  
Krzysztof Piersiala ◽  
...  

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