Juvenile-onset Respiratory Papillomatosis Research Articles

Intralesional Bevacizumab as Adjuvant Therapy for Juvenile Onset Recurrent Respiratory Papillomatosis: A Systematic Review.

Background: Respiratory papillomatosis is one of the common benign lesions of the airway that is often difficult to treat and carries significant morbidity. Bevacizumab is a monoclonal antibody that acts upon vascular endothelial growth factor receptor and is known to have an effect in respiratory papillomatosis. This study aims to systematically review the literature on efficacy of intralesional Bevacizumab in juvenile onset respiratory papillomatosis. Materials and methods. A systematic search of literature in various databases was conducted. The search was restricted to the English language, however, no restrictions were made regarding the date of publication keeping December 31st, 2020 as the last date of publication. We strictly complied with the PRISMA guidelines. Results. Of 145 articles analyzed, only 3 were selected as eligible and a total of twenty-one cases were evaluated. There was improvement in anatomic Derkay score after initiating intralesional Bevacizumb with reduction in the number of surgeries. Where reported, voice related functional outcomes also were also improved. No adverse effect related to the drug was reported. Conclusion: Intralesional Bevacizumab can be a promising efficacious, and safe adjuvant in the management of JORRP. Well-designed studies are further required in the future to prove its efficacy and safety over other adjuvants available.

Australian Paediatric Surveillance Unit (APSU) Annual Surveillance Report 2021.

The Australian Paediatric Surveillance Unit (APSU) has been conducting surveillance of rare communicable and non-communicable conditions in children since its inception in 1993. In this report, the results are described of surveillance of ten communicable diseases (and complications) for 2021, including the numbers of cases and incidence estimates; demographics; clinical features; and management and short-term outcomes. The included diseases are: acute flaccid paralysis (AFP); congenital cytomegalovirus (CMV); neonatal herpes simplex virus (HSV) infection; paediatric human immunodeficiency virus (HIV) infection; perinatal exposure to HIV; severe complications from influenza; juvenile-onset respiratory papillomatosis (JoRRP); congenital rubella syndrome; congenital varicella syndrome; and neonatal varicella infection. In 2021, cases of JoRRP were reported to the APSU for the first time since 2017, indicating potential gaps in HPV vaccination. AFP surveillance by APSU again contributed to Australia achieving a minimum target incidence of one AFP case per 100,000 children aged < 15 years. There were no cases of children with severe complications of influenza. No cases of varicella or congenital rubella were reported; however, at-risk populations, especially young migrant and refugee women from countries without universal vaccination programs, need to be screened and prioritised for vaccination prior to pregnancy. Cases of perinatal exposure to HIV continue to increase; however, the rate of mother-to-child-transmission remains at low levels due to the use of effective intervention strategies. Case numbers of congenital CMV and neonatal HSV remain steady in the absence of vaccines, prompting the need for greater awareness and education, with recent calls for target screening of at-risk infants for congenital CMV.

High recurrence rate in patients with juvenile-onset respiratory papillomatosis and its risk factors.

To identify the recurrence rate and risk factors for recurrence in patients with juvenile-onset recurrent respiratory papillomatosis (JORRP). A retrospective review was performed for all JORRP patients who underwent surgery between 2002 and 2019 at our institution. The demographic characteristics and clinical parameters were recorded. Kaplan-Meier estimates and Cox proportional hazards models were used to analyze the rate of recurrence and its risk factors. Our study included 721 patients. The cumulative recurrence rates at 1, 5, and 10 postoperative years following initial surgery were 74.2%, 90.0%, and 94.3%, respectively. Age at diagnosis younger than 4.5years (HR = 2.380, 95% CI [1.169-4.846], P = 0.017), high Derkay anatomical score (HR = 1.136, 95% CI [1.043-1.236], P = 0.003) and HPV type 11 infection (HR = 2.947, 95% CI [1.326-6.551], P = 0.008) were independent risk factors for recurrence. Adjuvant therapy with interferon was less likely to recur (HR = 0.237, 95% CI [0.091-0.616], P = 0.003). Additionally, gender, tracheotomy, mode of delivery, parity, expression of Ki-67, HPV vaccination, and surgical treatment method were not independently associated with recurrence (P > 0.05). Age at diagnosis younger than 4.5years, high Derkay anatomical score and HPV type 11 infection were associated with an increased risk for recurrence in patients with JORRP. Adjuvant therapy with interferon may reduce the risk of recurrence.

Human papillomavirus types causing recurrent respiratory papillomatosis in Zimbabwe

ObjectiveRecurrent respiratory papillomatosis (RRP) caused by human papillomavirus (HPV) is preventable through vaccination. This study was motivated by the recent thrust of the Zimbabwean government to reduce incidence of HPV related cervical cancer in Zimbabwe through vaccination against HPV. We therefore set out to type HPV genotypes causing RRP in Zimbabwe. We also describe for the first time, the demographics of Zimbabwean RRP patients, the characteristics of patients with different HPV types and possible risk factors of HPV infection in our setting. MethodsWe conducted a prospective, hospital based study were patients were recruited from two national otorhinolaryngology hospitals in Zimbabwe. All patients diagnosed with RRP during a twenty four month period were included in the study. A questionnaire was administered per patient to collect both demographic and clinical variables. HPV DNA was extracted from formalin fixed paraffin embedded laryngeal tissue. The extracted HPV DNA was amplified using polymerase chain reaction and next generation sequencing was used to genotype the HPV types. ResultsA total of 52 patients all aged 14 years and under were recruited into the study. Only Juvenile onset RRP cases were observed over the two year period and 64% of the patients were HPV positive. HPV types 6 and 11 were the dominant types observed constituting 85% of all HPV types. The remaining 15% constituted of HPV 16 and HPV 18. 27% of the patients had coinfection with at least two different HPV types. There were no statistically significant differences between the characteristics of HPV positive and HPV negative patients. No statistically significant risk factors were observed. ConclusionHPV types 6 and 11 were the predominant genotypes causing RRP in Zimbabwe. Thus the use of quadrivalent or even nonavalent HPV vaccines may play an important role in the prevention and management of RRP in Zimbabwe.

Clinical Significance of Low 2-Methoxyestradiol Levels in Serum and Tissue of Recurrent Juvenile-Onset Laryngeal Papillomatosis.

Objective We aim to explore the correlation between serum and tissue 2-methoxyestradiol (2-ME-2) levels and recurrence of juvenile-onset respiratory papillomatosis (JORRP). Study Design Retrospective cohort studies. Settings Laboratory of Otolaryngology, Department of Head and Neck Surgery, Renji Hospital, School of Medicine, Shanghai Jiaotong University. Subjects and Methods Sixty-four patients diagnosed with JORRP in our department from January 2007 to December 2012 were enrolled. Patients were divided into recurrence and nonrecurrence groups, with 32 patients in each group. ELISA detected the concentration of 2-ME-2 in serum and tissue samples collected during the first surgical procedure. Mann-Whitney analysis, receiver operating characteristic curves, logistic regression model, and Kaplan-Meier method were used for data processing. Results There was no difference in the serum 2-ME-2 concentration between the groups ( P = .237), while the tissue 2-ME-2 concentration of the recurrent group was significantly lower than that of the nonrecurrence group ( P = .0001). When the area under the curve was 0.752, the cutoff value of tissue 2-ME-2 at 670.02 pg/mL yielded the highest predictive sensitivity (71.9%) and specificity (71.9%). Regrouped by this cutoff point, patients with a lower tissue 2-ME-2 level (n = 26) had shorter disease-free survival and a higher recurrence odds ratio than patients with a higher tissue 2-ME-2 level (n = 38; P = .0408, odds ratio = 7.667). Conclusion A low tissue 2-ME-2 level is associated with a higher recurrence rate of JORRP. Tissue 2-ME-2 may be an effective target for JORRP treatment and a convenient measure for recurrence monitoring.

Juvenile-Onset Respiratory Papillomatosis: Caused by Maternal Condyloma in Pregnancy?

Respiratory papillomatosis, the most common benign neoplasm of the larynx in children, causes hoarseness, voice change, chronic cough, choking,

HLA class II polymorphisms and susceptibility to recurrent respiratory papillomatosis.

Recurrent respiratory papillomatosis (RRP) is characterised by multiple laryngeal papillomas. Left untreated, the lesions enlarge, spread, and endanger the airway. Medical treatments are unsatisfactory, and repeated surgery remains the mainstay of therapy. RRP is caused by human papillomavirus (HPV) infection. However, since oral HPV infection is common and RRP is rare, other host and/or viral factors may contribute to pathogenesis. In an attempt to identify such factors, we have investigated 60 patients. The patients were HLA class I, II, and tumor necrosis factor TNF typed by sequence-specific primer PCR, and the results compared to those for 554 healthy controls by using Fisher's exact test. Peripheral blood mononuclear cell proliferative responses of 25 controls and 10 patients to HPV-11 L1 virus-like particles (VLP) were compared. Short-term VLP-specific T-cell lines were established, and recognition of L1 was analyzed. Finally, the L1 open reading frames of HPV isolates from four patients were sequenced. Susceptibility to RRP was associated with HLA DRB1*0301 (33 of 60 patients versus 136 of 554 controls, P < 0.0001). The three most severely affected patients were homozygous for this allele. A range of T-cell proliferative responses to HPV-11 VLP were observed in DRB1*0301-positive healthy donors which were comparable to those in DRB1*0301-negative controls. Individuals with juvenile-onset RRP also mounted a range of VLP responses, and their magnitude was negatively correlated with the clinical staging score (P = 0.012 by the Spearman rank correlation). DRB1*0301-positive patients who responded to L1 recognized the same epitope as did matched controls and produced similar cytokines. Sequencing of clinical isolates excluded the possibility that nonresponsiveness was the result of mutation(s) in L1.