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      Pegylated interferon for treating severe recurrent respiratory papillomatosis in a child: case report

      case-report

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          ABSTRACT

          CONTEXT:

          Recurrent respiratory papillomatosis (RRP) is the most common laryngeal tumor. During childhood, it may present in extremely severe forms defined by the need for frequent surgical procedures to relieve respiratory distress and/or involvement of extralaryngeal sites such as lung involvement. Adjuvant therapies are indicated in these cases and interferon is one of the options. Pegylated interferon is more effective than conventional alpha interferon and, given its reported results in relation to treating hepatitis C over the past decade, we hypothesized that this might be more effective than conventional interferon also for treating respiratory papillomatosis. Use of a treatment strategy that eliminates the need for general anesthesia is particularly appealing, yet obtaining approval for use of medications that are not currently used for this purpose is challenging.

          CASE REPORT:

          We report the case of a child with severe RRP that had been followed for the preceding six years, who was treated with pegylated interferon after failure of other adjuvant therapies. There was noticeable improvement in the frequency of surgical procedures, which was regarded very receptively, considering the child’s history and previous response to other therapies.

          CONCLUSION:

          Pegylated interferon may be a good option for diminishing the need for surgical intervention in severe cases of recurrent respiratory papillomatosis.

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          Most cited references14

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          Peginterferon alfa-2b plus ribavirin compared with interferon alfa-2b plus ribavirin for initial treatment of chronic hepatitis C: a randomised trial.

          A sustained virological response (SVR) rate of 41% has been achieved with interferon alfa-2b plus ribavirin therapy of chronic hepatitis C. In this randomised trial, peginterferon alfa-2b plus ribavirin was compared with interferon alfa-2b plus ribavirin. 1530 patients with chronic hepatitis C were assigned interferon alfa-2b (3 MU subcutaneously three times per week) plus ribavirin 1000-1200 mg/day orally, peginterferon alfa-2b 1.5 microg/kg each week plus 800 mg/day ribavirin, or peginterferon alfa-2b 1.5 microg/kg per week for 4 weeks then 0.5 microg/kg per week plus ribavirin 1000-1200 mg/day for 48 weeks. The primary endpoint was the SVR rate (undetectable hepatitis C virus [HCV] RNA in serum at 24-week follow-up). Analyses were based on patients who received at least one dose of study medication. The SVR rate was significantly higher (p=0.01 for both comparisons) in the higher-dose peginterferon group (274/511 [54%]) than in the lower-dose peginterferon (244/514 [47%]) or interferon (235/505 [47%]) groups. Among patients with HCV genotype 1 infection, the corresponding SVR rates were 42% (145/348), 34% (118/349), and 33% (114/343). The rate for patients with genotype 2 and 3 infections was about 80% for all treatment groups. Secondary analyses identified bodyweight as an important predictor of SVR, prompting comparison of the interferon regimens after adjusting ribavirin for bodyweight (mg/kg). Side-effect profiles were similar between the treatment groups. In patients with chronic hepatitis C, the most effective therapy is the combination of peginterferon alfa-2b 1.5 microg/kg per week plus ribavirin. The benefit is mostly achieved in patients with HCV genotype 1 infections.
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            Recurrent respiratory papillomatosis: a review.

            Recurrent respiratory papillomatosis (RRP), which is caused by human papillomavirus types 6 and 11, is the most common benign neoplasm of the larynx among children and the second most frequent cause of childhood hoarseness. After changes in voice, stridor is the second most common symptom, first inspiratory and then biphasic. Less common presenting symptoms include chronic cough, recurrent pneumonia, failure to thrive, dyspnea, dysphagia, or acute respiratory distress, especially in infants with an upper respiratory tract infection. Differential diagnoses include asthma, croup, allergies, vocal nodules, or bronchitis. Reports estimate the incidence of RRP in the United States at 4.3 per 100,000 children and 1.8 per 100,000 adults. Infection in children has been associated with vertical transmission during vaginal delivery from an infected mother. Younger age at diagnosis is associated with more aggressive disease and the need for more frequent surgical procedures to decrease the airway burden. When surgical therapy is needed more frequently than four times in 12 months or there is evidence of RRP outside the larynx, adjuvant medical therapy should be considered. Adjuvant therapies that have been investigated include dietary supplements, control of extra-esophageal reflux disease, potent antiviral and chemotherapeutic agents, and photodynamic therapies; although several have shown promise, none to date has "cured" RRP, and some may have serious side effects. Because RRP, although histologically benign, is so difficult to control and can cause severe morbidity and death, better therapies are needed. The potential for a quadrivalent human papilloma vaccine is being explored to reduce the incidence of this disease.
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              Task force on recurrent respiratory papillomas. A preliminary report.

              C Derkay (1995)
              To obtain pilot data about the incidence, need for surgical intervention, and demographics of recurrent respiratory papillomas in the United States. Otolaryngologists were surveyed using a questionnaire with structured and open-ended questions. One thousand board-certified otolaryngologists practicing in the United States as of January 1, 1993, through a random mailing list provided by the American Academy of Otolaryngology-Head and Neck Surgery and all active US members of the American Society of Pediatric Otolaryngology and the American Bronchoesophagological Association. A total of 1346 questionnaires were distributed. Physician's responses to questions about their current patient load of children and adults with recurrent respiratory papillomas, their surgical and anesthetic management of the disease, and their clinical experiences with risk factors for developing recurrent respiratory papillomas. Projected totals for recurrent respiratory papillomas among children were 2354 new cases (95% confidence interval [CI], 1448 to 3260) and 5970 active cases (95% CI, 3465 to 8474), requiring 16,597 surgical procedures (95% CI, 6938 to 26,255) at a cost of $109 million (95% CI, $45 to 172 million) for March 1, 1993 to March 31, 1994. Projected totals for adult recurrent respiratory papillomas were 3623 new cases (95% CI, 2359 to 4887) and 9015 active cases (95% CI, 6435 to 11,591) requiring 9284 surgical procedures (95% CI, 6003 to 12,565) at a cost of $42 million (95% CI, $27 to $59 million) for March 1, 1993 to March 31, 1994. The incidence of recurrent respiratory papillomas among children is estimated at 4.3 per 100,000; among adults, 1.8 per 100,000. The carbon dioxide laser was favored by 92% of respondents. No consensus was reached on the role for cesarean section. Forty-six percent of respondents favored the use of a laser-safe endotracheal tube for anesthetic management. A registry of patients with recurrent respiratory papillomas would benefit future research protocols and provide long-term follow-up of patients.
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                Author and article information

                Journal
                Sao Paulo Med J
                Sao Paulo Med J
                Sao Paulo Med J
                São Paulo Medical Journal
                Associação Paulista de Medicina - APM
                1516-3180
                1806-9460
                18 September 2017
                2018
                : 136
                : 4
                : 376-381
                Affiliations
                [I ] originalMedical Doctor and Associate Professor, Department of Otolaryngology, Universidade Estadual de Campinas (UNICAMP), Campinas (SP), Brazil.
                [II ] originalMedical Doctor and Associate Professor, Department of Pediatrics, Universidade Estadual de Campinas (UNICAMP), Campinas (SP), Brazil.
                Author notes
                Address for correspondence: Rebecca Maunsell. Departamento de Otorrinolaringologia, Universidade Estadual de Campinas (UNICAMP). Rua Tessália Vieira de Camargo, 126, Campinas (SP) - Brasil. CEP 13083-887. Tel/Fax. (+55 19) 3521-7523. E-mail: rebecca.maunsell@ 123456gmail.com

                Conflict of interest: None

                Article
                10.1590/1516-3180.2017.0031240317
                9881702
                28977096
                bae56955-8497-409e-bb2c-2c2173d88215
                © 2022 by Associação Paulista de Medicina

                This is an open access article distributed under the terms of the Creative Commons license.

                History
                : 02 February 2017
                : 20 March 2017
                : 24 March 2017
                Page count
                Figures: 5, Tables: 2, Equations: 0, References: 12, Pages: 6
                Categories
                Case Report

                papillomatosis,respiratory,peginterferon,child
                papillomatosis, respiratory, peginterferon, child

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