Osseous sarcoidosis: A multicenter retrospective case-control study of 48 patients - 22/08/19

Doi : 10.1016/j.jbspin.2019.07.009

Imen Ben Hassine ^a,^d,^{^{1
Co-first author.}}, Christopher Rein ^e,^{^{1
Co-first author.}}, Cloé Comarmond ^a,^b,^c,^d,^{^{1
Co-first author.}}, Camille Glanowski ^f, Nathalie Saidenberg-Kermanac’h ^g, Benoît Meunier ^h, Nicolas Schleinitz ^h, Noémie Chanson ⁱ, Karim Sacré ⁱ, Marc Scherlinger ^j, Christophe Richez ^j, Sandrine Hirschi ^k, Matthieu Groh ^l, Hervé Devilliers ^m, Philip Bielefeld ^m, David Saadoun ^a,^b,^c,^d, Catherine Chapelon-Abric ^d, Laurent Arnaud ^n,^{^{2
Co-senior author.}}, Patrice Cacoub ^a,^b,^c,^d,^{^{2
Co-senior author.},}⁎
^a Sorbonne Universités, UPMC Univ Paris 06, UMR 7211, and Inflammation-Immunopathology-Biotherapy Department (DHU i2B), 75005 Paris, France
^b INSERM, UMR_S 959, 75013 Paris, France
^c CNRS, FRE3632, 75005 Paris, France
^d AP–HP, Groupe Hospitalier Pitié-Salpêtrière, Department of Internal Medicine and Clinical Immunology, 75013 Paris, France
^e Service de Médecine Interne et Rhumatologie, HIA Legouest, Metz, France
^f Hôpital d’instruction des armées de Bégin, Saint-Mandé, France
^g AP-HP, Groupe Hospitalier Avicenne-Jean Verdier-René Muret, Service de Rhumatologie, et Sorbonne Paris Cité, Université Paris 13, INSERM U1125, Bobigny, France
^h Service de Médecine Interne, CHU La Timone, Aix-Marseille Université, Marseille, France
ⁱ Service de Médecine Interne, CHU Bichat, Paris, France
^j Service de Rhumatologie, CHU de Bordeaux, Bordeaux, France
^k Service de Pneumologie, CHU de Strasbourg, Strasbourg, France
^l Service de Médecine Interne, CH Foch, Suresnes, France
^m Service de Médecine Interne, CHU Dijon Bourgogne, Dijon, France
ⁿ Service de Rhumatologie, Hôpitaux Universitaires de Strasbourg, Centre de Référence RESO, INSERM UMR-S1109, Université de Strasbourg, 67000 Strasbourg, France

^⁎Corresponding author. Department of Internal Medicine and Clinical Immunology, Hôpital La Pitié-Salpêtrière, 47-83, boulevard de l’Hôpital, 75651 Paris cedex 13, France.Department of Internal Medicine and Clinical Immunology, Hôpital La Pitié-Salpêtrière47-83, boulevard de l’HôpitalParis cedex 1375651France

Sous presse. Épreuves corrigées par l'auteur. Disponible en ligne depuis le Thursday 22 August 2019
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Highlights

•	Osseous sarcoidosis is frequently asymptomatic and incidentally detects in half of the patients.
•	Patients with osseous sarcoidosis have higher rates of thoracic and extra-thoracic lymph nodes, pulmonary and cutaneous involvement, and hypercalcemia.
•	Therapeutic response to corticosteroids is good in combination with methotrexate or hydroxychloroquin.

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Abstract

Objective

To describe the clinical presentation, distribution of lesions, treatment, and outcomes of osseous sarcoidosis.

Methods

A French retrospective multicenter study of patients with biopsy-proven sarcoidosis analyzed patients with 1) a biopsy-proven granuloma without caseous necrosis, and either 2) osseous clinical manifestations, or 3) abnormal osseous imaging. Sarcoidosis patients with osseous involvement (cases) were compared with 264 age- and sex-matched sarcoidosis patients with no osseous manifestations (controls).

Results

In the osseous sarcoidosis group (n=88), forty-two (48%) patients had osseous-related symptoms involving the axial (69%) and/or appendicular (58%) skeleton. On imaging, the most commonly affected bones were in the spine (52%), pelvis (42%), hands (22%) and femur (19%). Compared with controls, cases had higher rates of mediastinal (93% vs. 47%) and extra-thoracic lymph node involvement (66% vs. 21%), pulmonary (90% vs. 65%) and cutaneous involvement (44% vs. 23%) (all P<0.0001), and hypercalcemia (8.5% vs. 2%, P=0.014). Spleen/liver and gastrointestinal involvement were less frequent in the osseous sarcoidosis group (29% vs. 45%, and 1% vs. 17%, respectively, P<0.0001). Response rates to with glucocorticoids alone, glucocorticoids plus methotrexate or glucocorticoids plus hydroxychloroquine were 23/44 (52%), 9/13 (69%) and 4/6 (67%), respectively.

Conclusion

In patients with osseous sarcoidosis the spine and pelvis were the most commonly affected bones. Compared with controls, cases with osseous sarcoidosis have higher rates of thoracic and extra-thoracic lymph node involvement, pulmonary and cutaneous involvement, and hypercalcemia. Most patients with osseous sarcoidosis had a good response to glucocorticoids in combination with methotrexate or hydroxychloroquine.

Le texte complet de cet article est disponible en PDF.

Keywords : Sarcoidosis, Osseous manifestations, Bone involvement