Literature DB >> 28064338

Nasal closure for severe hereditary haemorrhagic telangiectasia in 100 patients. The Lund modification of the Young's procedure: a 22-year experience.

V J Lund1, Y Darby1, J Rimmer2, M Amin3, S Husain4.   

Abstract

INTRODUCTION: Hereditary haemorrhagic telangiectasia (HHT) is an autosomal dominant vascular disease characterized by recurrent epistaxis, mucocutaneous telangiectasia and visceral arteriovenous malformations. The nosebleeds can be life-threatening and in these circumstances, radical treatment is required.
METHODS: Since 1994, closure of the nose has been undertaken to prevent severe nasal bleeding in patients meeting specific selection criteria. Outcome data collected on this cohort pre- and post-operatively is available for analysis.
RESULTS: From a cohort of 515 HHT patients, 100 have undergone nasal closure, bilateral ab initio in 81%. Fifty patients completed pre- and post-operative epistaxis severity questionnaires and provided information on post-operative problems and 28 completed a Glasgow Benefit Inventory (GBI). Overall most patients derived significant benefit from the procedure with complete cessation of nasal bleeding in 94%, a highly significant improvement in the epistaxis score and a mean GBI score of 53.4. Loss of smell and taste was the most frequent post-operative complaint.
CONCLUSION: HHT can be associated with life-threatening epistaxis significantly affecting patients quality of life. Permanent prevention of airflow is associated with complete or near-total cessation of epistaxis in the majority of patients.

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Year:  2017        PMID: 28064338     DOI: 10.4193/Rhin16.315

Source DB:  PubMed          Journal:  Rhinology        ISSN: 0300-0729            Impact factor:   3.681


  6 in total

1.  Low serum haptoglobin and blood films suggest intravascular hemolysis contributes to severe anemia in hereditary hemorrhagic telangiectasia.

Authors:  Lieze Thielemans; D Mark Layton; Claire L Shovlin
Journal:  Haematologica       Date:  2018-10-18       Impact factor: 9.941

2.  Patient-recorded benefit from nasal closure in a Danish cohort of patients with hereditary haemorrhagic telangiectasia.

Authors:  Jonas Hjelm Andersen; Anette Drøhse Kjeldsen
Journal:  Eur Arch Otorhinolaryngol       Date:  2019-12-16       Impact factor: 2.503

3.  Mutational and phenotypic characterization of hereditary hemorrhagic telangiectasia.

Authors:  Claire L Shovlin; Ilenia Simeoni; Kate Downes; Zoe C Frazer; Karyn Megy; Maria E Bernabeu-Herrero; Abigail Shurr; Jennifer Brimley; Dilipkumar Patel; Loren Kell; Jonathan Stephens; Isobel G Turbin; Micheala A Aldred; Christopher J Penkett; Willem H Ouwehand; Luca Jovine; Ernest Turro
Journal:  Blood       Date:  2020-10-22       Impact factor: 22.113

4.  European Reference Network For Rare Vascular Diseases (VASCERN) Outcome Measures For Hereditary Haemorrhagic Telangiectasia (HHT).

Authors:  Claire L Shovlin; Elisabetta Buscarini; Anette D Kjeldsen; Hans Jurgen Mager; Carlo Sabba; Freya Droege; Urban Geisthoff; Sara Ugolini; Sophie Dupuis-Girod
Journal:  Orphanet J Rare Dis       Date:  2018-08-15       Impact factor: 4.123

5.  The effects of nasal closure on quality of life in patients with hereditary hemorrhagic telangiectasia.

Authors:  Rhett S Thomson; Nicole L Molin; Kevin J Whitehead; Shaelene Ashby; Leland Johnson; P Daniel Ward; Bryan R McRae; Kevin F Wilson; Jamie McDonald
Journal:  Laryngoscope Investig Otolaryngol       Date:  2018-04-19

6.  Endoscopic surgical treatment of epistaxis in hereditary haemorrhagic telangiectasia: our experience.

Authors:  Fabio Pagella; Alessandro Pusateri; Eugenia Maiorano; Giuseppe Spinozzi; Sara Ugolini; Roberta Lizzio; Rosolino Mirabella; Carmine Tinelli; Carla Olivieri; Elina Matti
Journal:  Acta Otorhinolaryngol Ital       Date:  2021-02       Impact factor: 2.124

  6 in total

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