Research & Literature

Anaphylaxis to anakinra in a pediatric patient with systemic juvenile idiopathic arthritis successfully treated with canakinumab: a case-based review.

Aguiar CL, Pan N, Adams A, et al.

Clinical rheumatology 2015; (34(10)):1821-4 doi:10.1007/s10067-015-2889-y.

Tolerance and efficacy of off-label anti-interleukin-1 treatments in France: a nationwide survey.

Rossi-Semerano L, Fautrel B, Wendling D, et al.

Orphanet journal of rare diseases 2015; (10()):19 doi:10.1186/s13023-015-0228-7.

Chronic recurrent multifocal osteomyelitis: a rare skeletal disorder.

Aygun D, Barut K, Camcioglu Y, Kasapcopur O

BMJ case reports 2015; (2015()).

Anakinra-Induced Acute Liver Failure in an Adolescent Patient with Still's Disease.

Taylor SA, Vittorio JM, Martinez M, et al.

Pharmacotherapy 2016; (36(1)):e1-4 doi:10.1002/phar.1677.

Lipin-2 regulates NLRP3 inflammasome by affecting P2X7 receptor activation.

Lordén G, Sanjuán-García I, de Pablo N, et al.

The Journal of experimental medicine 2017; (214(2)):511-528 doi:10.1084/jem.20161452.

Deficiency of Interleukin-1 Receptor Antagonist (DIRA): Report of the First Indian Patient and a Novel Deletion Affecting IL1RN.

Mendonca LO, Malle L, Donovan FX, et al.

Journal of clinical immunology 2017; (37(5)):445-451 doi:10.1007/s10875-017-0399-1.

Bone metabolism and inflammatory characteristics in 14 cases of chronic nonbacterial osteomyelitis.

Ata Y, Inaba Y, Choe H, et al.

Pediatric rheumatology online journal 2017; (15(1)):56 doi:10.1186/s12969-017-0183-z.

Dealing with Chronic Non-Bacterial Osteomyelitis: a practical approach.

Taddio A, Ferrara G, Insalaco A, et al.

Pediatric rheumatology online journal 2017; (15(1)):87 doi:10.1186/s12969-017-0216-7.

Bone inflammation and chronic recurrent multifocal osteomyelitis.

Chen Z, Cheng L, Feng G

European review for medical and pharmacological sciences 2018; (22(5)):1380-1386 doi:10.26355/eurrev_201803_14482.

Deficiency of Interleukin-1 Receptor Antagonist: A Case with Late Onset Severe Inflammatory Arthritis, Nail Psoriasis with Onychomycosis and Well Responsive to Adalimumab Therapy.

Kutukculer N, Puel A, Eren Akarcan S, et al.

Case reports in immunology 2019; (2019()):1902817 doi:10.1155/2019/1902817.

Compound heterozygous LPIN2 pathogenic variants in a patient with Majeed syndrome with recurrent fever and severe neutropenia: case report.

Liu J, Hu XY, Zhao ZP, et al.

BMC medical genetics 2019; (20(1)):182 doi:10.1186/s12881-019-0919-3.

Chronic recurrent multifocal osteomyelitis: a case report.

Açoğlu EA, Akçaboy M, Yıldız YT, et al.

Turk pediatri arsivi 2019; (54(4)):272-276 doi:10.14744/TurkPediatriArs.2018.69379.

Whole-body MRI in the diagnosis of paediatric CNO/CRMO.

Andronikou S, Kraft JK, Offiah AC, et al.

Rheumatology (Oxford, England) 2020; (59(10)):2671-2680 doi:10.1093/rheumatology/keaa303.

Comparing children and adults with chronic nonbacterial osteomyelitis.

Lenert A, Ferguson PJ

Current opinion in rheumatology 2020; (32(5)):421-426 doi:10.1097/BOR.0000000000000734.

Novel Majeed Syndrome-Causing LPIN2 Mutations Link Bone Inflammation to Inflammatory M2 Macrophages and Accelerated Osteoclastogenesis.

Bhuyan F, de Jesus AA, Mitchell J, et al.

Arthritis & rheumatology (Hoboken, N.J.) 2021; (73(6)):1021-1032 doi:10.1002/art.41624.

Majeed Syndrome: A Review of the Clinical, Genetic and Immunologic Features.

Ferguson PJ, El-Shanti H

Biomolecules 2021; (11(3)) doi:10.3390/biom11030367.

Chronic nonbacterial osteomyelitis (CNO) and chronic recurrent multifocal osteomyelitis (CRMO).

Zhao DY, McCann L, Hahn G, Hedrich CM

Journal of translational autoimmunity 2021; (4()):100095 doi:10.1016/j.jtauto.2021.100095.

Majeed Syndrome: Five Cases With Novel Mutations From Unrelated Families in India With a Review of Literature.

Chavan PP, Aksentijevich I, Daftary A, et al.

The Journal of rheumatology 2021; (48(12)):1850-1855 doi:10.3899/jrheum.201663.

Patient-reported effectiveness and safety of Pamidronate in NSAIDs-refractory chronic recurrent multifocal osteomyelitis in children.

Juszczak B, Sułko J

Rheumatology international 2022; (42(4)):699-706 doi:10.1007/s00296-021-04886-4.

[Clinical and genetic analysis of a child with Majeed syndrome].

Sun L, Zhang P, Song Y, et al.

Zhonghua yi xue yi chuan xue za zhi = Zhonghua yixue yichuanxue zazhi = Chinese journal of medical genetics 2021; (38(8)):775-778 doi:10.3760/cma.j.cn511374-20200612-00432.

Chronic Nonbacterial Osteomyelitis: Insights into Pathogenesis, Assessment, and Treatment.

Nuruzzaman F, Zhao Y, Ferguson PJ

Rheumatic diseases clinics of North America 2021; (47(4)):691-705 doi:10.1016/j.rdc.2021.06.005.

Neurosurgical management of vertebral lesions in pediatric chronic recurrent multifocal osteomyelitis: patient series.

Hug NF, Purger DA, Li D, et al.

Journal of neurosurgery. Case lessons 2023; (5(4)).

Juvenile idiopathic arthritis with systemic onset with inflammatory bone lesions: two case reports of patients successfully treated with canakinumab and experience gained from literature.

Alexeeva EI, Dvoryakovskaya TM, Tsulukiya IT, et al.

Frontiers in pediatrics 2023; (11()):1163483 doi:10.3389/fped.2023.1163483.

Anakinra and hepatotoxicity in pediatric rheumatology: a case series.

Martins FR, Azevedo AC, Ganhão S, et al.

Pediatric rheumatology online journal 2023; (21(1)):112 doi:10.1186/s12969-023-00891-y.

LPIN2 -related Majeed syndrome: report of two Indian patients with novel variants in LPIN2 and review of literature.

Badiger VA, Balan S, Madan S, et al.

Clinical dysmorphology 2024; (33(1)):27-30 doi:10.1097/MCD.0000000000000476.

Chronic Recurrent Multifocal Osteomyelitis.

Rafferty BA, Thakrar P

The Medical clinics of North America 2024; (108(1)):227-239 doi:10.1016/j.mcna.2023.05.022.

Safety and efficacy of canakinumab treatment for undifferentiated autoinflammatory diseases: the data of a retrospective cohort two-centered study.

Alexeeva E, Shingarova M, Dvoryakovskaya T, et al.

Frontiers in medicine 2023; (10()):1257045 doi:10.3389/fmed.2023.1257045.

Whole-Body MRI at Initial Presentation of Chronic Recurrent Multifocal Osteomyelitis, Juvenile Idiopathic Arthritis, Their Overlapping Syndrome, and Non-Specific Arthropathy.

Lanckoroński M, Gietka P, Mańczak M, Sudoł-Szopińska I

Journal of clinical medicine 2024; (13(4)) doi:10.3390/jcm13040998.

Spine Involvement and Vertebral Deformity in Patients Diagnosed with Chronic Recurrent Multifocal Osteomyelitis.

Rogers ND, Trizno AA, Joyce CD, et al.

Journal of pediatric orthopedics 2024; (44(9)):561-566 doi:10.1097/BPO.0000000000002743.

Majeed syndrome: first description in a patient of central-European ancestry.

Drago E, Bertoni A, Grossi A, et al.

Rheumatology (Oxford, England) 2025; (64(5)):3069-3073 doi:10.1093/rheumatology/keae480.

Chronic Recurrent Multifocal Osteomyelitis Involving the Spine, Sternum, and Lower Extremities: A Case Report.

Huynh K, McLendon L, Woolnough L, Elder ME

Pediatrics 2024; (154(4)) doi:10.1542/peds.2024-067527.

Autoinflammatory Bone Diseases.

Haşlak F, Akay N, Gül Ü, et al.

Balkan medical journal 2025; (42(1)):5-13 doi:10.4274/balkanmedj.galenos.2024.2024-11-129.

Sterile osteomyelitis: a cardinal sign of autoinflammation.

Borges T, Santos J, Silva S

Reumatologia 2024; (62(6)):475-488 doi:10.5114/reum/196595.

Surgical Interventions in Chronic Recurrent Multifocal Osteomyelitis Affecting the Spine: A Case Report with Literature Review.

Lovaković J, Banić T, Ehrenfreund T, et al.

Journal of orthopaedic case reports 2025; (15(3)):93-99 doi:10.13107/jocr.2025.v15.i03.5346.

Chronic Non-Bacterial Osteomyelitis (CNO) in a Tertiary Center in Southern Italy: Response to Treatment and Outcome Stratification.

Loconte R, Donghia R, Francavilla M, et al.

Children (Basel, Switzerland) 2025; (12(4)) doi:10.3390/children12040451.

Post-marketing safety of anakinra and canakinumab: a real-world pharmacovigilance study based on FDA adverse event reporting system.

Liu H, Yan W, Li J, et al.

Frontiers in pharmacology 2025; (16()):1483669 doi:10.3389/fphar.2025.1483669.

Retrospective Review of 80 Patients with Chronic Recurrent Multifocal Osteomyelitis Evaluated by Pediatric Orthopaedic Surgeons.

Taylor TN, Bridges CS, Ezeokoli EU, et al.

Journal of the Pediatric Orthopaedic Society of North America 2023; (5(1)):575 doi:10.55275/JPOSNA-2023-575.

Childhood Leukemia Presenting as Clinical Arthritis and Chronic Recurrent Multifocal Osteomyelitis: A Case Report.

Al Masroori E, Al Hadhrami S, Al Shamli N

Oman medical journal 2025; (40(2)):e743 doi:10.5001/omj.2025.12.

Vertebral fractures and bone health in patients with chronic non-bacterial osteomyelitis at disease onset: insights from the monocentric CAMELOT cohort.

Chighizola CB, Di Taranto R, Amati A, et al.

Therapeutic advances in musculoskeletal disease 2025; (17()):1759720X251375183 doi:10.1177/1759720X251375183.

Clinical and genetic analysis of Majeed syndrome caused by LPIN2 complex heterozygous mutation and literature review.

Wang S, Yu T, He X, et al.

Frontiers in pediatrics 2025; (13()):1602509 doi:10.3389/fped.2025.1602509.

Cutaneous manifestations of autoinflammatory bone diseases: a retrospective multicenter study.

Öner N, Cüceoğlu MK, Könte EK, et al.

Clinical rheumatology 2026; (45(2)):1245-1256 doi:10.1007/s10067-025-07870-6.

Chronic nonbacterial osteomyelitis: current perspectives in pediatric practice.

Başaran Ö, Yıldız AE, Bilginer Y, Özen S

The Turkish journal of pediatrics 2026; (68(1)):1-17 doi:10.24953/turkjpediatr.2026.7367.

Diagnosis, treatment and monitoring of chronic nonbacterial osteomyelitis (CNO) and chronic recurrent multifocal osteomyelitis (CRMO) - Evidence, practice and consensus-based recommendations from the German pediatric rheumatology society (GKJR).

Schnabel A, Reiser C, Beer M, et al.

Autoimmunity reviews 2026; (25(7)):104075 doi:10.1016/j.autrev.2026.104075.

Metaphyseal changes on whole-body magnetic resonance imaging following bisphosphonate therapy in children with chronic nonbacterial osteomyelitis: distinguishing treatment effect from disease flare.

Karmazyn B, Kleppel H, Chilukuri SR, et al.

Pediatric radiology 2026; doi:10.1007/s00247-026-06819-4.