Successful Treatment of an Advanced Larynx Carcinoma Using Neo-Adjuvant Chemo-Immunotherapy and Cisplatin/Radiotherapy for a Patient with Rendu–Osler Disease

<b>Background/Objectives</b>: Rendu-Osler disease is a rare genetic disease, characterized by widespread telangiectasia that can involve the skin and mucous membranes. The diagnosis is based on spontaneous and recurrent epistaxis; various mucosal and cutaneous telangiectasia at typical sites; visceral manifestations including gastrointestinal telangiectasia or pulmonary, cerebral, or hepatic arteriovenous malformation; and a first-degree relative with hereditary hemorrhagic telangiectasia. Squamous cell carcinoma of the larynx generally develops in patients with a smoking history. It is most often treated by surgery and/or radiotherapy. To our knowledge, these two entities were never reported in the same patient. <b>Methods:</b> We herein report a case of a 51-year-old man with Rendu-Osler disease. He was subsequently diagnosed with a locally advanced well-differentiated squamous cell carcinoma of the vocal cord. <b>Results:</b> The patient received a neo-adjuvant chemo-immunotherapy, with nine weekly injections of paclitaxel (60 mg/m<sup>2</sup>/week), cisplatin (30 mg/m<sup>2</sup>/week), and cetuximab (250 mg/m<sup>2</sup>/week), and three injections of pembrolizumab (200 mg every 3 weeks). This controlled tumor bleeding, and then cisplatin-enhanced radiotherapy helped obtain a complete remission. <b>Conclusions:</b> Locally advanced squamous cell carcinoma of the larynx treatment in the context of active Rendu-Osler disease is challenging. If the wide curative surgical approach is deemed too risky, neo-adjuvant chemo-immunotherapy may present a helpful alternative as it may enhance the conditions in order to perform classical radiotherapy with concomitant cisplatin.

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Journal
Journal of Clinical Medicine
Published
2025-08-12
DOI
https://doi.org/10.3390/jcm14165694
Primary Topic
Tracheal and airway disorders
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article
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article

Successful Treatment of an Advanced Larynx Carcinoma Using Neo-Adjuvant Chemo-Immunotherapy and Cisplatin/Radiotherapy for a Patient with Rendu–Osler Disease

Tamim Alsuliman, Abdenour Ouikene, Farid Belkhir, T Garnier et al.
Journal of Clinical Medicine
Tracheal and airway disorders
article

Successful Treatment of an Advanced Larynx Carcinoma Using Neo-Adjuvant Chemo-Immunotherapy and Cisplatin/Radiotherapy for a Patient with Rendu–Osler Disease

Tamim Alsuliman, Abdenour Ouikene, Farid Belkhir, T Garnier, Alessandro Cau, Réda Garidi, Agnes Galez, V. Salle, Hanene Oueslati, Bruno Chauffert, Sana Nemmaoui
article en

Abstract

<b>Background/Objectives</b>: Rendu-Osler disease is a rare genetic disease, characterized by widespread telangiectasia that can involve the skin and mucous membranes. The diagnosis is based on spontaneous and recurrent epistaxis; various mucosal and cutaneous telangiectasia at typical sites; visceral manifestations including gastrointestinal telangiectasia or pulmonary, cerebral, or hepatic arteriovenous malformation; and a first-degree relative with hereditary hemorrhagic telangiectasia. Squamous cell carcinoma of the larynx generally develops in patients with a smoking history. It is most often treated by surgery and/or radiotherapy. To our knowledge, these two entities were never reported in the same patient. <b>Methods:</b> We herein report a case of a 51-year-old man with Rendu-Osler disease. He was subsequently diagnosed with a locally advanced well-differentiated squamous cell carcinoma of the vocal cord. <b>Results:</b> The patient received a neo-adjuvant chemo-immunotherapy, with nine weekly injections of paclitaxel (60 mg/m<sup>2</sup>/week), cisplatin (30 mg/m<sup>2</sup>/week), and cetuximab (250 mg/m<sup>2</sup>/week), and three injections of pembrolizumab (200 mg every 3 weeks). This controlled tumor bleeding, and then cisplatin-enhanced radiotherapy helped obtain a complete remission. <b>Conclusions:</b> Locally advanced squamous cell carcinoma of the larynx treatment in the context of active Rendu-Osler disease is challenging. If the wide curative surgical approach is deemed too risky, neo-adjuvant chemo-immunotherapy may present a helpful alternative as it may enhance the conditions in order to perform classical radiotherapy with concomitant cisplatin.

Journal of Clinical MedicineVol. 14(16)
Centre Hospitalier de Saint-Quentin (FR), Centre Hospitalier Universitaire Amiens-Picardie (FR)
Openalex Percentile: Top 69%
Tracheal and airway disorders
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