Pulmonary manifestations of chronic HPV infection in patients with recurrent respiratory papillomatosis

Pai, Sara I.; Wasserman, Isaac; Ji, Yisi D.; Gilman, Matthew D.; Hung, Yin P.; Faquin, William C.; Mino‐Kenudson, Mari; Muniappan, Ashok

doi:10.1016/s2213-2600(22)00008-x

Cited by 4 publications

(4 citation statements)

References 57 publications

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“…Specifically, recent reviews focusing on individuals aged less than 20 years demonstrated an estimated incidence of lung involvement equal to 3.3% and an estimated incidence of lung tumor of 16% in the same category of patients [ 18 ]. In agreement with our results, Pai and Colleagues (2022) reported an incidence of 8.9% of pulmonary involvement in the RRP population, with a higher risk in younger patients, in those undergoing multiple surgical operations, in those tracheostomized, and in those with tracheal involvement [ 14 ].…”

Section: Discussionsupporting

confidence: 92%

“…Although the anatomical sites infected by HPV are mainly larynx and trachea, malignant transformation in bronchi and lungs (<1% of all lung neoplasms) can rarely occur, representing the primary cause of death in this patient population [ 9 , 13 , 14 , 15 , 16 ]. Malignancy, which can occur after surgical/chemotherapeutic therapies or after a relapse, within decades from disease onset [ 17 , 18 , 19 ], is the consequence of translocation of fragments from the larynx to the lung tissue after therapeutic manipulations [ 20 , 21 ].…”

Section: Introductionmentioning

confidence: 99%

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Pulmonary Involvement in Recurrent Respiratory Papillomatosis: A Systematic Review

Sechi,

Muresu,

Di Lorenzo

et al. 2024

Infectious Disease Reports

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Recurrent respiratory papillomatosis (RRP) is a non-malignant disease, characterized by the production of wart-like growths in the respiratory tract, affecting both young people and adults (juvenile-onset recurrent respiratory papillomatosis, JORRP, and adult-onset recurrent respiratory papillomatosis, AORRP, respectively). Infection caused by human papillomavirus (HPV) is known as the main factor involved in RRP development. Complications of RRP may rarely occur, including lung involvement and malignant transformation. The present systematic review aimed to evaluate the prevalence of severe complications, such as lung involvement and lung tumor in JORRP and AORRP patients, and assess the role of HPV genotypes in the progression of disease severity following the guideline for reporting systematic reviews and meta-analysis (PRISMA Statement). A total of 378 studies were found on PubMed and Scopus using the following MESH terms: “recurrent respiratory papillomatosis and lung tumor” and “pulmonary tumor and recurrent respiratory papillomatosis”. Basing on inclusion and exclusion criteria, a total of 11 studies were included in the systematic review. We found a pooled prevalence of 8% (95% CI: 4–14%; I2: 87.5%) for lung involvement in RRP patients. In addition, we found a pooled risk difference of 5% in lung involvement between JORRP and AORRP (95% CI: −7–18%; I2: 85.6%, p-value: 0.41). Among patients with lung involvement, we observed a pooled prevalence of lung tumor of 4% (95% CI:1–7%; I2: 67.1%) and a pooled prevalence mortality for this group of 4% (95% CI:2–6%; I2: 0%). Overall, the positivity rate for HPV-6 and -11 in patients with RRP was 91%. Considering only cases with pulmonary involvement, the pooled prevalence for HPV-11 was 21% (95% CI: 5–45%; I2: 77.2%). Our results evidenced a low/middle risk of pulmonary involvement and lung tumor in JORRP and AORRP patients, with an increased risk for HPV-11-positive patients. Further studies should be performed to improve knowledge and adopt preventive measures to contrast the progression to severe diseases in RRP patients.

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Section: Discussionsupporting

confidence: 92%

Section: Introductionmentioning

confidence: 99%

Pulmonary Involvement in Recurrent Respiratory Papillomatosis: A Systematic Review

Sechi,

Muresu,

Di Lorenzo

et al. 2024

Infectious Disease Reports

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“…A vascular endothelial growth factor binding antibody, systemic bevacizumab has been shown in several case reports to prolong papilloma-free intervals and thereby decrease the number of surgical interventions needed, all while presenting a relatively low complication profile. [5][6][7][8][9][10][11][12][13][14][15][16][17] As pulmonary extension represents the most refractory, morbid manifestation of RRP and presents risk for malignant transformation, 18 understanding the efficacy of systemic bevacizumab for this RRP sub-population is an important clinical need. To date, however, efficacy studies of systemic bevacizumab for pulmonary RRP are limited largely by their single-institutional natures and small sample sizes.…”

Section: Introductionmentioning

confidence: 99%

“…As pulmonary extension represents the most refractory, morbid manifestation of RRP and presents risk for malignant transformation, 18 understanding the efficacy of systemic bevacizumab for this RRP sub‐population is an important clinical need. To date, however, efficacy studies of systemic bevacizumab for pulmonary RRP are limited largely by their single‐institutional natures and small sample sizes 5,7,10,19 .…”

Section: Introductionmentioning

confidence: 99%

Efficacy of Systemic Bevacizumab for Recurrent Respiratory Papillomatosis with Pulmonary Involvement

Lopez

Ballestas

et al. 2023

The Laryngoscope

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ObjectivesPulmonary papillomatosis is a rare but severe manifestation of recurrent respiratory papillomatosis (RRP). Efficacy data of systemic bevacizumab for pulmonary RRP are limited. This study's objective was to characterize disease response of pulmonary RRP to systemic bevacizumab.MethodsA retrospective review was performed to identify patients with pulmonary RRP seen at three medical institutions. Clinical symptoms, CT findings, and disease response were compared before and after initiation of systemic bevacizumab therapy. Disease response was categorized as complete response, partial response, stabilization, or progression for each subsite involved by papilloma.ResultsOf the 12 pulmonary RRP patients treated with systemic bevacizumab, 4 (33.3%) were male, and 11 (91.7%) were juvenile‐onset RRP patients. All presented with laryngeal, tracheal, and pulmonary RRP. The median (range) age at first bevacizumab infusion was 48.1 (19.5–70.2) years. Progression to pulmonary malignancy was identified in 3 (25.0%) patients, 2 before initiation of and 1 after complete cessation of bevacizumab therapy. Clinical symptoms such as dyspnea (75.0% vs. 25.0%; p = 0.01) and dysphagia and/or odynophagia (33.3 vs. 0.0%; p = 0.03) were significantly decreased following bevacizumab therapy. Compared with pre‐treatment baseline, 9 (75.0%) patients experienced a stable‐to‐partial response in the lungs to systemic bevacizumab, and 10 (83.3%) experienced partial‐to‐complete responses in the larynx and trachea.ConclusionSystemic bevacizumab is effective in stabilizing progression in even the most severe cases of RRP, with both a dramatic reduction in laryngeal and tracheal disease as well as a stable‐to‐partial response of pulmonary involvement in a majority of patients.Level of Evidence4 Laryngoscope, 2023

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Systemic Bevacizumab for Recurrent Respiratory Papillomatosis: A Single Institution's Experience

So,

Rayle,

Joo

et al. 2024

The Laryngoscope

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ObjectivesMedical therapies to limit disease recurrence are critically needed for recurrent respiratory papillomatosis (RRP). Systemic bevacizumab is emerging as an exciting adjuvant therapy toward this end, but uptake has been poor due to the lack of experience and awareness of best prescribing practices. The objective of this study was to describe a single tertiary care academic medical center's experience using systemic bevacizumab for the treatment of RRP.MethodsA retrospective review was performed to identify patients with RRP on systemic bevacizumab. Demographic and clinical characteristics, findings on imaging reports, and disease response at all anatomic subsites involved in papilloma were documented.ResultsOf the 17 RRP patients on systemic bevacizumab, 9 (52.9%) were male, and 12 (70.6%) were diagnosed with juvenile‐onset RRP. The total lifetime number of surgeries was high, with more than half (n = 9; 52.9%) undergoing more than 50 surgeries. Following induction of systemic bevacizumab, a significant reduction in patients with laryngeal (n = 15; 94.1% vs. n = 7; 41.2%, p < 0.001) and tracheal (n = 11; 64.7% vs. n = 5; 29.4%, p = 0.04) RRP was noted. Surgical frequency was significantly lower following systemic bevacizumab (2.5 vs. 0.5 surgeries per year; p < 0.001). The most common complications were new‐onset hypertension (n = 4; 23.5%) and proteinuria (n = 5; 29.4%).ConclusionSystemic bevacizumab is effective in reducing the number of surgeries needed for RRP while exhibiting a relatively safe complication profile. Papillomas in the larynx and trachea are most responsive to systemic bevacizumab, while pulmonary RRP is most likely to exhibit a partial‐to‐stable response.Level of Evidence4 Laryngoscope, 2024

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Pulmonary manifestations of chronic HPV infection in patients with recurrent respiratory papillomatosis

Cited by 4 publications

References 57 publications

Pulmonary Involvement in Recurrent Respiratory Papillomatosis: A Systematic Review

Pulmonary Involvement in Recurrent Respiratory Papillomatosis: A Systematic Review

Efficacy of Systemic Bevacizumab for Recurrent Respiratory Papillomatosis with Pulmonary Involvement

Systemic Bevacizumab for Recurrent Respiratory Papillomatosis: A Single Institution's Experience

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