In inflammatory myopathies, dropped head/bent spine syndrome is associated with scleromyositis: an international case-control study.


Journal

RMD open
ISSN: 2056-5933
Titre abrégé: RMD Open
Pays: England
ID NLM: 101662038

Informations de publication

Date de publication:
09 2023
Historique:
received: 18 02 2023
accepted: 22 06 2023
medline: 6 9 2023
pubmed: 5 9 2023
entrez: 4 9 2023
Statut: ppublish

Résumé

Some myopathies can lead to dropped head or bent spine syndrome (DH/BS). The significance of this symptom has not been studied in inflammatory myopathies (IM). To assess the significance of DH/BS in patients with IM. Practitioners from five IM networks were invited to report patients with IM suffering from DH/BS (without other known cause than IM). IM patients without DH/BS, randomly selected in each participating centre, were included as controls at a ratio of 2 to 1. 49 DH/BS-IM patients (DH: 57.1%, BS: 42.9%) were compared with 98 control-IM patients. DH/BS-IM patients were older (65 years vs 53 years, p<0.0001) and the diagnosis of IM was delayed (6 months vs 3 months, p=0.009). Weakness prevailing in the upper limbs (42.9% vs 15.3%), dysphagia (57.1% vs 25.5%), muscle atrophy (65.3% vs 34.7%), weight loss (61.2% vs 23.5%) and loss of the ability to walk (24.5% vs 5.1%) were hallmarks of DH/BS-IM (p≤0.0005), for which the patients more frequently received intravenous immunoglobulins (65.3% vs 34.7%, p=0.0004). Moreover, DH/BS-IM patients frequently featured signs and/or complications of systemic sclerosis (SSc), fulfilling the American College of Rheumatology/European Alliance of Associations for Rheumatology criteria for this disease in 40.8% of the cases (vs 5.1%, p<0.0001). Distribution of the myopathy, its severity and its association with SSc were independently associated with DH/BS (p<0.05). Mortality was higher in the DH/BS-IM patients and loss of walking ability was independently associated with survival (p<0.05). In IM patients, DH/BS is a marker of severity and is associated with SSc (scleromyositis).

Sections du résumé

BACKGROUND
Some myopathies can lead to dropped head or bent spine syndrome (DH/BS). The significance of this symptom has not been studied in inflammatory myopathies (IM).
OBJECTIVES
To assess the significance of DH/BS in patients with IM.
METHODS
Practitioners from five IM networks were invited to report patients with IM suffering from DH/BS (without other known cause than IM). IM patients without DH/BS, randomly selected in each participating centre, were included as controls at a ratio of 2 to 1.
RESULTS
49 DH/BS-IM patients (DH: 57.1%, BS: 42.9%) were compared with 98 control-IM patients. DH/BS-IM patients were older (65 years vs 53 years, p<0.0001) and the diagnosis of IM was delayed (6 months vs 3 months, p=0.009). Weakness prevailing in the upper limbs (42.9% vs 15.3%), dysphagia (57.1% vs 25.5%), muscle atrophy (65.3% vs 34.7%), weight loss (61.2% vs 23.5%) and loss of the ability to walk (24.5% vs 5.1%) were hallmarks of DH/BS-IM (p≤0.0005), for which the patients more frequently received intravenous immunoglobulins (65.3% vs 34.7%, p=0.0004). Moreover, DH/BS-IM patients frequently featured signs and/or complications of systemic sclerosis (SSc), fulfilling the American College of Rheumatology/European Alliance of Associations for Rheumatology criteria for this disease in 40.8% of the cases (vs 5.1%, p<0.0001). Distribution of the myopathy, its severity and its association with SSc were independently associated with DH/BS (p<0.05). Mortality was higher in the DH/BS-IM patients and loss of walking ability was independently associated with survival (p<0.05).
CONCLUSION
In IM patients, DH/BS is a marker of severity and is associated with SSc (scleromyositis).

Identifiants

pubmed: 37666644
pii: rmdopen-2023-003081
doi: 10.1136/rmdopen-2023-003081
pmc: PMC10481849
pii:
doi:

Types de publication

Journal Article

Langues

eng

Sous-ensembles de citation

IM

Commentaires et corrections

Type : ErratumIn

Informations de copyright

© Author(s) (or their employer(s)) 2023. Re-use permitted under CC BY-NC. No commercial re-use. See rights and permissions. Published by BMJ.

Déclaration de conflit d'intérêts

Competing interests: None declared.

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Auteurs

Luc Pijnenburg (L)

Service de rhumatologie, Centre de Référence des Maladies Auto-immunes Systémiques Rares Est Sud-Ouest Hôpitaux Universitaires de Strasbourg, Université de Strasbourg, Strasbourg, France.

Margherita Giannini (M)

Service de rhumatologie, Centre de Référence des Maladies Auto-immunes Systémiques Rares Est Sud-Ouest Hôpitaux Universitaires de Strasbourg, Université de Strasbourg, Strasbourg, France.
Exploration fonctionnelle musculaire, Service de Physiologie Explorations Fonctionnelles Musculaires, Hôpitaux Universitaires de Strasbourg, Université de Strasbourg, Strasbourg, France.

Maude Bouchard-Marmen (M)

Division of Rheumatology, CHU de Quebec-Universite Laval, Quebec, Quebec, Canada.

Laurent Arnaud (L)

Service de rhumatologie, Centre de Référence des Maladies Auto-immunes Systémiques Rares Est Sud-Ouest Hôpitaux Universitaires de Strasbourg, Université de Strasbourg, Strasbourg, France.

Simone Barsotti (S)

Department of Rheumatology and Clinical Immunology Unit, University of Pisa, Pisa, Italy.

Silvia Bellando-Randone (S)

Department of Experimental and Clinical Medicine, Universitaria Careggi, Azienda Ospedaliera, Florence, Italy.

Livio Bernardi (L)

Departement of rheumatology, University of Padova, Padova, Italy.

Paola Bini (P)

Department of neurology, Neurological Institute Mondino, University of Pavia, Pavia, Italy.

Jelena Blagojevic (J)

Department of Experimental and Clinical Medicine, Universitaria Careggi, Azienda Ospedaliera, Florence, Italy.

Veronica Codullo (V)

Rheumatology Division, University and IRCCS Policlinico S. Matteo Foundation Pavia, Pavia, Italy.

Marion Couderc (M)

Service de rhumatologie, Hôpitaux Universitaires de Clermont-Ferrand, Clermont-Ferrand, France.

Claire De Moreuil (C)

Service de médecine interne, Hôpitaux Universitaires de Brest, Brest, France.

Emanuelle Dernis (E)

Service de rhumatologie, Centre hospitalier Le Mans, Le Mans, France.

Luca Diamanti (L)

Department of neurology, Neurological Institute Mondino, University of Pavia, Pavia, Italy.

Jean Jacques Dubost (JJ)

Service de médecine interne, Hôpitaux Universitaires de Clermont-Ferrand, Clermont-Ferrand, France.

Fanny Duval (F)

Service de neurologie, Hôpitaux Universitaires de Bordeaux, Bordeaux, France.

Giacomo Emmi (G)

Department of Experimental and Clinical Medicine, Universitaria Careggi, Azienda Ospedaliera, Florence, Italy.

Jean-Marc Galempoix (JM)

Service de Médecine Interne, Centre Hospitalier de Charleville-Mézières, Charleville-Mézières, France.

Bernard Geny (B)

Exploration fonctionnelle musculaire, Service de Physiologie Explorations Fonctionnelles Musculaires, Hôpitaux Universitaires de Strasbourg, Université de Strasbourg, Strasbourg, France.

Jacques-Eric Gottenberg (JE)

Service de rhumatologie, Centre de Référence des Maladies Auto-immunes Systémiques Rares Est Sud-Ouest Hôpitaux Universitaires de Strasbourg, Université de Strasbourg, Strasbourg, France.

Monica Groza (M)

Service de médecine interne, Centre hospitalier Colmar, Colmar, France.

Aurelien Guffroy (A)

Service d'immunologie clinique, Centre de Référence des Maladies Auto-immunes Systémiques Rares Est Sud-Ouest Hôpitaux Universitaires de Strasbourg, Université de Strasbourg, Strasbourg, France.

Isabelle Guichard (I)

Service de médecine interne, Hôpitaux Universitaires de Saint Etienne, Saint Etienne, France.

Philippe Guilpain (P)

Service de médecine interne, Hôpitaux Universitaires de Montpellierr, Montpellier, France.

Baptiste Hervier (B)

Service de médecine interne, Assistance publique - Hôpitaux de Paris, Saint Louis, Paris, France.

Marie Hudson (M)

Department of medicine, Division of Rheumatology, Jewish General Hospital, McGill University, Montreal, Québec, Canada.

Luca Iaccarino (L)

Departement of rheumatology, University of Padova, Padova, Italy.

Florenzo Iannone (F)

Department of rheumatology, University of Bari, Bari, Italy.

Delphine Lebrun (D)

Service de Médecine Interne, Centre Hospitalier de Charleville-Mézières, Charleville-Mézières, France.

Enrico Marchioni (E)

Department of neurology, Neurological Institute Mondino, University of Pavia, Pavia, Italy.

Kuberaka Mariampillai (K)

Service de médecine interne, Assistance publique - Hôpitaux de Paris, Saint Louis, Paris, France.

Francois Maurier (F)

Service de médecine interne, Hôpitaux privés de Metz, Metz, France.

Marta Mosca (M)

Department of Rheumatology and Clinical Immunology Unit, University of Pisa, Pisa, Italy.

Aleksandra Nadaj-Pakleza (A)

Service de neurologie, Centre de référence des maladies neuromusculaires, Hôpitaux Universitaires de Strasbourg, Strasbourg, France.

Carlotta Nannini (C)

Department of Rheumatology, Prato Hospital, Prato, Italy.

Jean-Maxime Piot (JM)

Service de rhumatologie, Centre hospitalier Le Mans, Le Mans, France.

Sergio Prieto-González (S)

Department of internal medicine, Hospital Clinic of Barcelona, Barcelona, Spain.

Nicolas Poursac (N)

Service de rhumatologie, Centre de Référence des Maladies Auto-immunes Systémiques Rares Est Sud-Ouest, Hôpitaux Universitaires de Bordeaux, Bordeaux, France.

Eglantine Rouanet (E)

Service de rhumatologie, Centre hospitalier Le Mans, Le Mans, France.

Jérémie Sellam (J)

Service de rhumatologie, Hôpital Saint Antoine, Assistance publique - Hôpitaux de Paris, Paris, France.

Albert Selva-O'Callaghan (A)

Department of internal medicine, Val d'Hebron General Hospital, Barcelona, Spain.

François Séverac (F)

Service de Santé Publique, GMRC; ICube, UMR 7357, Hôpitaux universitaires de Strasbourg, Université de Strasbourg, Strasbourg, France.

Jean Sibilia (J)

Service de rhumatologie, Centre de Référence des Maladies Auto-immunes Systémiques Rares Est Sud-Ouest Hôpitaux Universitaires de Strasbourg, Université de Strasbourg, Strasbourg, France.

Guilhem Sole (G)

Service de neurologie, Hôpitaux Universitaires de Bordeaux, Bordeaux, France.

Antoine Soulages (A)

Service de neurologie, Hôpitaux Universitaires de Bordeaux, Bordeaux, France.

Benjamin Terrier (B)

Service de médecine interne, Hôpital Cochin Assistance publique - Hôpitaux de Paris Cochin, Paris, France.

Anne Tournadre (A)

Service de rhumatologie, Hôpitaux Universitaires de Clermont-Ferrand, Clermont-Ferrand, France.

Yves Troyanov (Y)

Department of rheumatology, Hôpital Sacre Cœur Montréal, Université de Montréal, Montreal, Québec, Canada.

Nathalie Vernier (N)

Service de médecine interne, Hôpitaux Universitaires de Dijon, Dijon, France.

Veronique Vesperini (V)

Service de rhumatologie, Centre hospitalier Narbonne, Narbonne, France.

Jean-François Viallard (JF)

Service de médecine interne, Hôpitaux Universitaires de Bordeaux, Bordeaux, France.

Rahima Ziane (R)

Service de médecine interne, Hôpitaux Universitaires de Montpellierr, Montpellier, France.

Lorenzo Cavagna (L)

Rheumatology Division, University and IRCCS Policlinico S. Matteo Foundation Pavia, Pavia, Italy.

Alain Meyer (A)

Exploration fonctionnelle musculaire, Service de Physiologie Explorations Fonctionnelles Musculaires, Hôpitaux Universitaires de Strasbourg, Université de Strasbourg, Strasbourg, France alain.meyer1@chru-strasbourg.fr.

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