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Introduction Recurrent respiratory papillomatosis (RRP) is a chronic disease caused by human papillomavirus (HPV), characterized by recurrent papillomas in the respiratory tract. Presenting as either juvenile-onset RRP (JoRRP) or adult-onset RRP (AoRRP), the severity of the disease is subjective and unpredictable. Lack of curative therapies necessitates disease management involving repeated surgical removal of lesions. The review aimed to assess the clinical, humanistic and economic burden associated with RRP. Methods Systematic literature reviews of Embase ® , MEDLINE ® and Cochrane databases were conducted for epidemiology, clinical, humanistic, and economic burden, from database inception to November 30, 2022. Conference abstracts were also searched (2019–2022). Key inclusion criteria consisted of juveniles or adults with RRP/laryngeal papillomatosis, with no restriction on study country, interventions, or comparators. Outcomes of interest included incidence, prevalence, risk factors, symptomatic presentation, HPV genotype, cost burden, resource use and health related quality of life (HRQoL). Results In JoRRP, the incidence rate ranged from 0.2–2.1 per 100,000 and the prevalence rate ranged from 0.8–4.3 per 100,000. Incidence and prevalence of AoRRP were 0.2–3.9 and 0.4–8.4 per 100,000, respectively. Limited studies reported the subsequent impact of introducing national prophylactic HPV immunisation programs on JoRRP epidemiology, but where available, they were associated with significantly reduced incidence rates. Symptomatic presentations were diverse, with voice impact and breathing difficulties commonly reported. More aggressive disease was linked to earlier age of onset and HPV11 genotype. Healthcare utilisation was largely driven by surgical interventions, due to lack of curative treatments. Cost burden was substantial, with JoRRP associated with triple the costs of AoRRP in the US. Patients with JoRRP and AoRRP experienced considerable HRQoL impairment, particularly relating to voice disorder. Conclusion Extensive clinical, humanistic and economic disease burden was reported for both JoRRP and AoRRP, as it is a chronic condition, with propensity to recur and spread. Feasibility of improving HPV prophylactic vaccination coverage against HPV6/HPV11 should be explored to reduce incidence, alongside efforts to improve treatment of JoRRP and AoRRP patients. Despite the existing literature, RRP remains a poorly understood disease, and future research on risk factors and medical options are needed. Supplementary Information The online version contains supplementary material available at 10.1186/s12931-024-03057-w.
Introduction Recurrent respiratory papillomatosis (RRP) is a chronic disease caused by human papillomavirus (HPV), characterized by recurrent papillomas in the respiratory tract. Presenting as either juvenile-onset RRP (JoRRP) or adult-onset RRP (AoRRP), the severity of the disease is subjective and unpredictable. Lack of curative therapies necessitates disease management involving repeated surgical removal of lesions. The review aimed to assess the clinical, humanistic and economic burden associated with RRP. Methods Systematic literature reviews of Embase ® , MEDLINE ® and Cochrane databases were conducted for epidemiology, clinical, humanistic, and economic burden, from database inception to November 30, 2022. Conference abstracts were also searched (2019–2022). Key inclusion criteria consisted of juveniles or adults with RRP/laryngeal papillomatosis, with no restriction on study country, interventions, or comparators. Outcomes of interest included incidence, prevalence, risk factors, symptomatic presentation, HPV genotype, cost burden, resource use and health related quality of life (HRQoL). Results In JoRRP, the incidence rate ranged from 0.2–2.1 per 100,000 and the prevalence rate ranged from 0.8–4.3 per 100,000. Incidence and prevalence of AoRRP were 0.2–3.9 and 0.4–8.4 per 100,000, respectively. Limited studies reported the subsequent impact of introducing national prophylactic HPV immunisation programs on JoRRP epidemiology, but where available, they were associated with significantly reduced incidence rates. Symptomatic presentations were diverse, with voice impact and breathing difficulties commonly reported. More aggressive disease was linked to earlier age of onset and HPV11 genotype. Healthcare utilisation was largely driven by surgical interventions, due to lack of curative treatments. Cost burden was substantial, with JoRRP associated with triple the costs of AoRRP in the US. Patients with JoRRP and AoRRP experienced considerable HRQoL impairment, particularly relating to voice disorder. Conclusion Extensive clinical, humanistic and economic disease burden was reported for both JoRRP and AoRRP, as it is a chronic condition, with propensity to recur and spread. Feasibility of improving HPV prophylactic vaccination coverage against HPV6/HPV11 should be explored to reduce incidence, alongside efforts to improve treatment of JoRRP and AoRRP patients. Despite the existing literature, RRP remains a poorly understood disease, and future research on risk factors and medical options are needed. Supplementary Information The online version contains supplementary material available at 10.1186/s12931-024-03057-w.
Recurrent respiratory papillomatosis (RRP) is a rare, chronic disease caused by the human papilloma virus. A delay in appropriate intervention could be caused by the symptomatology being similar to bronchial asthma.A 5-year-old boy presented to the pediatric emergency unit of Usmanu Danfodiyo University Teaching Hospital with a 2-year history of recurrent difficulty in breathing, cough and noisy breathing. He had previously received several treatments for asthma at Primary Health Care (PHC) facilities. On examination, he was dyspneic with a respiratory rate of 60 cycles/minute, oxygen saturation was 86% on room air, and chest auscultation showed vesicular breath sounds. Initially, acute severe asthma was diagnosed. However, further examination revealed wart-like lesions protruding below the pharynx, and a diagnosis of respiratory papillomatosis was considered. He had an emergency tracheostomy and video-assisted laryngoscopy with excision of the lesions. The histological report confirmed a laryngeal papilloma. His clinical condition improved after removal of wart-like lesions, and he was discharged afterwards. He had three relapses and surgical removal (September and December 2023, June 2024). Currently, he is stable and is being followed up at the Pediatric and Otorhinolaryngology Clinic.
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