Table 1.
Cases of MRH (Multicentric reticulohistiocytosis) associated with autoimmune diseases reported in the literature up until August 2022. Data adapted from the following articles: (1) MRH associated with primary biliary cirrhosis: successful treatment with cytotoxic agents [21]; (2) MRH associated with subclinical Sjögren’s syndrome [22]; (3) MRH in a Taiwanese woman with Sjögren syndrome [23]; (4) MRH associated with RA (rheumatoid arthritis) [24]; (5) Paraneoplastic multicentric reticulohistiocytosis associated with a celiac disease [25]; (6) MRH: a systemic osteoclastic disease? [26]; (7) Case report of an association with erosive RA and systemic Sjogren syndrome [18]; (8) A Rare Case of MRH with Concurrent RA [27]; AIDs = autoimmune diseases; y.o. = years old; PBC = primary biliary cirrhosis; AI = autoimmune; RF = rheumatoid factor; CS = corticosteroids, MTX = methotrexate; TG = transglutaminases; DM = diabetes mellitus.
| Timing of Clinical Manifestations | AIDs Associated | Serology | Timing of Diagnosis | Therapy | |
|---|---|---|---|---|---|
| Case 1. Caucasian woman, 56 y.o. |
Joint manifestations preceded dermatological manifestations | Vitiligo, PBC, AI thyroiditis |
Positive thyroid microsomal and thyroglobulin antibodies | MRH diagnosis following AIDs diagnosis | NSAIDs, D-penicillamine, cyclophosphamide, chlorambucil |
| Case 2 Caucasian woman, 60 y.o. |
Joint manifestations preceded dermatological manifestations (7 years) | Sjogren syndrome | Positive ANA (1:80) and anti-Ro/SSA antibodies | Pre-existent Sjogren’s syndrome | Cyclophosphamide, CS, sodium aurothiomalate |
| Case 3 Taiwanese woman, 59 y.o. |
Joint manifestations preceded dermatological manifestations (2 years) |
Sjogren’s syndrome | Positive ANA (1:640), anti-Ro/SSA and anti-La/SSB | Pre-existent Sjogren’s syndrome | CS, hydroxychloroquine |
| Case 4 Japanese woman, 67 y.o. |
Joint manifestations preceded dermatological manifestations (20 years) | Seropositive RA | Positive RF | MRH diagnosis followed RA diagnosis (22 years later) | MTX, CS |
| Case 5 Caucasian woman, 68 y.o. |
- | Celiac disease | Positive anti-endomysium, anti-TG, and anti-gliadin | CS, MTX | |
| Case 6 Caucasian woman, 40 y.o. |
- | Sjogren’s syndrome, RA |
Positive RF and anti–CCP | Sjogren’s diagnoses preceded MRH diagnoses | CS, hydroxychloroquine, MTX, intravenous infusions of zoledronic acid |
| Case 7 Caucasian woman, 50 y.o. |
Concomitant | RA, Sjogren’s syndrome |
Positive RF, ACPA, and anti-Ro/SSA | MRH complicated by Sjogren’s syndrome | MTX, CS, alendronate |
| Case 8 Caucasian woman, 60 y.o. |
- | Type 1 DM | Anemia, elevated VES, PCR, RF, anti- CCP titer | - | NSAIDs, MTX, CS, hydroxychloroquine, ibandronate |