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<p>Glandular involvement of the head and neck is an extremely rare feature in ANCA-associated vasculitis (AAV). The objective was to describe glandular involvement in AAV by analysing data from a national cohort and comparing the findings to those from a systematic literature review. A multicentric retrospective study was conducted through a survey promoted by the <i>French Vasculitis Study Group</i> and included patients aged ≥ 16&#xa0;years who met the ACR/EULAR classification criteria for AAV and had lachrymal and/or salivary involvement. Demographic, clinical and biological findings were analysed and pooled with data from a MEDLINE review of the literature since inception until June 5, 2024. The study population included 20 patients with a median age of 52&#xa0;years (IQR, 44.5–57.5&#xa0;years). Granulomatosis with polyangiitis (<i>n</i> = 17) and PR3-ANCA (<i>n</i> = 10) were the most represented. Parotitis (<i>n</i> = 9) and dacryoadenitis (<i>n</i> = 8) were found to precede and/or were present at AAV onset and recurrences. All patients received glucocorticoids, usually in combination with immunosuppressants. The systematic review identified another 67 cases. Pooled analysis of all cases (<i>n</i> = 87) showed that salivary gland involvement was not only frequently associated with ENT features (OR = 4.01 (95% CI, 1.30–13.06; <i>p</i> &lt; 0.01)), but also with PR3-ANCA positivity (OR = 3.34 (95% CI, 1.09–11.34; <i>p</i> = 0.02)). Lachrymal involvement was associated with concomitant ophthalmological signs (OR = 3.93 (95% CI, 1.31–12.14; <i>p</i> &lt; 0.01)). Glandular involvement of the head and neck is a rare but identifiable manifestation of active AAV. Knowledge of this uncommon presentation refines clinical assessment for optimal management of patients with AAV.</p>

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Glandular Manifestations of the Head and Neck in ANCA-Associated Vasculitides: Data from a National Cohort and Systematic Review

  • 期刊论文

  • 发表日期：

    2025年10月15日

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  • Nicolas Duhamel,
  • Xavier Puéchal,
  • Thomas Le Gallou,
  • Julie Chezel,
  • Alban Deroux,
  • Céline Roussin,
  • Jérémy Keraen,
  • Claire De Moreuil,
  • Pierre-André Jarrot,
  • Mathieu Gerfaud-Valentin,
  • François Maurier,
  • Julien Campagne,
  • Isabelle Melki,
  • Sara Melboucy,
  • Sophie Nagle,
  • Benjamin Terrier,
  • Nihal Martis

摘要

Glandular involvement of the head and neck is an extremely rare feature in ANCA-associated vasculitis (AAV). The objective was to describe glandular involvement in AAV by analysing data from a national cohort and comparing the findings to those from a systematic literature review. A multicentric retrospective study was conducted through a survey promoted by the French Vasculitis Study Group and included patients aged ≥ 16 years who met the ACR/EULAR classification criteria for AAV and had lachrymal and/or salivary involvement. Demographic, clinical and biological findings were analysed and pooled with data from a MEDLINE review of the literature since inception until June 5, 2024. The study population included 20 patients with a median age of 52 years (IQR, 44.5–57.5 years). Granulomatosis with polyangiitis (n = 17) and PR3-ANCA (n = 10) were the most represented. Parotitis (n = 9) and dacryoadenitis (n = 8) were found to precede and/or were present at AAV onset and recurrences. All patients received glucocorticoids, usually in combination with immunosuppressants. The systematic review identified another 67 cases. Pooled analysis of all cases (n = 87) showed that salivary gland involvement was not only frequently associated with ENT features (OR = 4.01 (95% CI, 1.30–13.06; p < 0.01)), but also with PR3-ANCA positivity (OR = 3.34 (95% CI, 1.09–11.34; p = 0.02)). Lachrymal involvement was associated with concomitant ophthalmological signs (OR = 3.93 (95% CI, 1.31–12.14; p < 0.01)). Glandular involvement of the head and neck is a rare but identifiable manifestation of active AAV. Knowledge of this uncommon presentation refines clinical assessment for optimal management of patients with AAV.