Klinik Araştırma
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Chronic Nonbacterial Osteomyelitis in Children: A Single Center Experience

Yıl 2024, Cilt: 7 Sayı: 2, 169 - 174, 30.06.2024

Öz

Objective
Chronic nonbacterial osteomyelitis (CNO), an inflammatory bone disease, results from immune cell cytokine overproduction. Patients may complain of bone and joint pain, myalgia, and mucocutaneous lesions, impacting all bones. We aim to share clinical insights on CNO diagnosis, management, and monitoring.
Method
We retrospectively analyzed the demographic and clinical data of 9 children diagnosed with CNO at our pediatric rheumatology clinic. Patients aged 0-18 years who were followed for at least 6 months were included.
Results
Out of the 9 CNO patients, 3 (33.3%) were female and 6 (66.7%) were male. The median age at diagnosis was 11 years (range: 6.5-16 years), and the diagnosis delay averaged 14.3 months (range: 2-36 months). Fatigue and bone pain were the most common symptoms. In physical examination, localized tenderness was found in 7 (77.7%) patients, arthritis in 6 (66.6%), and bone deformity in 1 (11%). Magnetic resonance imaging revealed multiple bone lesions in 8 (88.8%) patients, primarily in the lower extremities. Non-steroidal anti-inflammatory treatment was initiated for all patients. Methotrexate was introduced in 7 (77.7%) patients during follow-up, and one patient received a biological agent due to multifocal lesions. Three patients transitioned from methotrexate to a biological agent due to inadequate clinical and laboratory improvement. Additionally, bisphosphonate was combined with methotrexate for one patient due to vertebral height reduction. Clinical remission was achieved in 8 patient.
Conclusion
As a result, the data we uncovered in our study regarding the clinical characteristics, radiological findings, treatment options, and responses to anti-inflammatory treatments in children diagnosed with CNO may raise awareness, especially among pediatricians. We predict that, with heightened physician awareness, the delay in diagnosis will decrease, and treatment success will increase.

Kaynakça

  • Jansson AF, Müller TH, Gliera L, et al. Clinical score for nonbacterial osteitis in children and adults. Arthritis Rheum. 2009;60(4):1152-1159. doi:10.1002/art.24402
  • Jansson A, Renner ED, Ramser J, et al. Classification of non-bacterial osteitis: Retrospective study of clinical, immunological and genetic aspects in 89 patients. Rheumatology. 2007;46(1):154-160. doi:10.1093/rheumatology/kel190
  • Wipff J, Costantino F, Lemelle I, et al. A large national cohort of French patients with chronic recurrent multifocal osteitis. Arthritis and Rheumatology. 2015;67(4):1128-1137. doi:10.1002/art.39013
  • Zhao Y, Wu EY, Oliver MS, et al. Consensus Treatment Plans for Chronic Nonbacterial Osteomyelitis Refractory to Nonsteroidal Antiinflammatory Drugs and/or With Active Spinal Lesions. Arthritis Care Res (Hoboken). 2018;70(8):1228-1237. doi:10.1002/acr.23462
  • Voit AM, Arnoldi AP, Douis H, et al. Whole-body magnetic resonance imaging in chronic recurrent multifocal osteomyelitis: Clinical longterm assessment may underestimate activity. Journal of Rheumatology. 2015;42(8):1455-1462. doi:10.3899/jrheum.141026
  • Beck C, Morbach H, Beer M, et al. Chronic nonbacterial osteomyelitis in childhood: Prospective follow-up during the first year of anti-inflammatory treatment. Arthritis Res Ther. 2010;12(2). doi:10.1186/ar2992
  • Morbach H, Hedrich CM, Beer M, Girschick HJ. Autoinflammatory bone disorders. Clinical Immunology. 2013;147(3):185-196. doi:10.1016/j.clim.2012.12.012
  • Tronconi E, Miniaci A, Baldazzi M, Greco L, Pession A. Biologic treatment for chronic recurrent multifocal osteomyelitis: report of four cases and review of the literature. Rheumatol Int. 2018;38(1):153-160. doi:10.1007/s00296-017-3877-0
  • Taddio A, Ferrara G, Insalaco A, et al. Dealing with Chronic Non-Bacterial Osteomyelitis: A practical approach. Pediatric Rheumatology. 2017;15(1). doi:10.1186/s12969-017-0216-7
  • De Vries E, Van Der Weij JP, Veen CJPVD, et al. In Vitro Effect of (3-Amino-l-Hydroxypropylidene)-1,1-Bisphosphomnc Acid (APD) on the Function of Mononuclear Phagocytes in Lymphocyte Proliferation.; 1982.
  • Santini D, Fratto ME, Vincenzi B, Cesa A La, Dianzani C, Tonini G. DRUG MECHANISMS AND TARGETS Bisphosphonate Effects in Cancer and Inflammatory Diseases In Vitro and In Vivo Modulation of Cytokine Activities. Vol 18.; 2004.
  • Girschick H, Finetti M, Orlando F, et al. The multifaceted presentation of chronic recurrent multifocal osteomyelitis: A series of 486 cases from the Eurofever international registry. Rheumatology (United Kingdom). 2018;57(7):1203-1211. doi:10.1093/rheumatology/key058
  • Sözeri B, Ayaz NA, Atıkan BY, et al. Clinical experiences in Turkish paediatric patients with chronic recurrent multifocal osteomyelitis. Turkish Journal of Pediatrics. 2019;61(6):879-884. doi:10.24953/turkjped.2019.06.009
  • Wipff J, Costantino F, Lemelle I, et al. A large national cohort of French patients with chronic recurrent multifocal osteitis. Arthritis and Rheumatology. 2015;67(4):1128-1137. doi:10.1002/art.39013
  • Sağ E, Sönmez HE, Demir S, et al. Chronic recurrent multifocal osteomyelitis in children: A single center experience over five years. Turkish Journal of Pediatrics. 2019;61(3):386-391. doi:10.24953/turkjped.2019.03.010
  • Khanna G, Sato TS, Ferguson P. Imaging of chronic recurrent multifocal osteomyelitis. Radiographics. 2009;29:1159-1177.
  • Hassan M, Assi H, Hassan M, et al. Chronic Recurrent Multifocal Osteomyelitis: A Comprehensive Literature Review. Cureus. Published online August 8, 2023. doi:10.7759/cureus.43118
  • Ulu K, İşgüder R, Karadağ ŞG, et al. Clinical characteristics and predictors for recurrence in chronic nonbacterial osteomyelitis: a retrospective multicenter analysis. Turk J Med Sci. 2023;53(5):1105-1111. doi:10.55730/1300-0144.5675
  • Zhao Y, Wu EY, Oliver MS, et al. Consensus Treatment Plans for Chronic Nonbacterial Osteomyelitis Refractory to Nonsteroidal Antiinflammatory Drugs and/or With Active Spinal Lesions. Arthritis Care Res (Hoboken). 2018;70(8):1228-1237. doi:10.1002/acr.23462

Çocuklarda Kronik Non Bakteriyel Osteomiyelit: Tek Merkez Deneyimi

Yıl 2024, Cilt: 7 Sayı: 2, 169 - 174, 30.06.2024

Öz

Giriş
Kronik non-bakteriyel osteomiyelit (KNO), bağışıklık hücreleri tarafından sitokinlerin aşırı ekspresyonunun neden olduğu inflamatuar bir hastalıktır. Hastalar kemik ağrısı, eklem ağrısı, miyalji, mukokutonöz lezyonlar ile hastaneye başvurabilmektedir. Hastalık tüm kemikleri tutabilme özelliğine sahiptir. Bu çalışmanın amacı KNO tanı, tedavi ve takip süreci ile ilgili klinik deneyimlerimizi paylaşmaktır.
Yöntem
Pediatrik romatoloji polikliniğinde KNO tanısı alan 9 çocuğun demografik verileri ve klinik özellikleri geriye dönük olarak incelendi. En az 6 ay takip edilen 0-18 yaş arası hastalar dahil edildi.
Bulgular
Çalışmaya dahil edilen 9 KNO hastasının 3’ü (%33,3) kız, 6’sı (%66,7) erkekti. Ortanca tanı yaşları 11 (6,5-16) yıl ve tanı gecikme süresi 14,3 (2-36) aydı. En sık başvuru bulgusu halsizlik ve kemik ağrısıydı. Fizik muayenede; 7 (%77,7) hastada lokalize hassasiyet, 6’ında (%66,6) artrit ve birinde (%11) kemik deformitesi saptandı. Manyetik rezonans görüntülemede, 8 (%88,8) hastada birden çok kemik lezyonu vardı. Hastaların 8’inde (%88,8) alt ekstremite tutulumu vardı ve klavikula tutulumu dışında hastaların hiçbirinde üst ekstremite tutulumu yoktu. Tüm hastalara steroid dışı anti-inflamatuar tedavi başlandı. İzlemde 7 (%77,7) hastaya metotreksat başlandı. Bir hastanın tedavisine çok odaklı tutulumu olduğundan biyolojik ajan eklendi. Metotreksat başlanan 7 hastadan 3’ünde (%33,3) yeterli klinik ve laboratuvar düzelme olmadığı için biyolojik ajana geçildi. Ayrıca 1 (%11) hastada vertebrada yükseklik kaybı olduğu için metotreksat tedavisine bifosfonat eklendi. Sekiz hasta klinik remisyona ulaştı.
Sonuç
Sonuç olarak çalışmamızda KNO tanısı alan çocukların klinik özellikleri, radyolojik bulguları, tedavi seçenekleri, anti-inflamatuar tedavilere yanıtları ile ilgili saptadığımız veriler özellikle çocuk hekimleri için farkındalık yaratabilir. Farkındalığı olan hekimler sayesinde tanı gecikmesinin azaltacağını, tedavi başarısının artıracağını öngörmekteyiz.

Etik Beyan

Kocaeli Üniversitesi Tıp Fakültesi Klinik Araştırmalar Etik Kurulu onayı alınmıştır. Tüm prosedürler, kurumsal ve/veya ulusal araştırma komitesinin etik standartlarına ve 1964 Helsinki Bildirgesi’ne uygun olarak gerçekleştirilmiştir. (Onay numarası ve tarihi: E-80418770-020-490041 sayılı 23.10.2023 evrak tarihli GOKAEK-2023/17.12)

Destekleyen Kurum

yoktur

Kaynakça

  • Jansson AF, Müller TH, Gliera L, et al. Clinical score for nonbacterial osteitis in children and adults. Arthritis Rheum. 2009;60(4):1152-1159. doi:10.1002/art.24402
  • Jansson A, Renner ED, Ramser J, et al. Classification of non-bacterial osteitis: Retrospective study of clinical, immunological and genetic aspects in 89 patients. Rheumatology. 2007;46(1):154-160. doi:10.1093/rheumatology/kel190
  • Wipff J, Costantino F, Lemelle I, et al. A large national cohort of French patients with chronic recurrent multifocal osteitis. Arthritis and Rheumatology. 2015;67(4):1128-1137. doi:10.1002/art.39013
  • Zhao Y, Wu EY, Oliver MS, et al. Consensus Treatment Plans for Chronic Nonbacterial Osteomyelitis Refractory to Nonsteroidal Antiinflammatory Drugs and/or With Active Spinal Lesions. Arthritis Care Res (Hoboken). 2018;70(8):1228-1237. doi:10.1002/acr.23462
  • Voit AM, Arnoldi AP, Douis H, et al. Whole-body magnetic resonance imaging in chronic recurrent multifocal osteomyelitis: Clinical longterm assessment may underestimate activity. Journal of Rheumatology. 2015;42(8):1455-1462. doi:10.3899/jrheum.141026
  • Beck C, Morbach H, Beer M, et al. Chronic nonbacterial osteomyelitis in childhood: Prospective follow-up during the first year of anti-inflammatory treatment. Arthritis Res Ther. 2010;12(2). doi:10.1186/ar2992
  • Morbach H, Hedrich CM, Beer M, Girschick HJ. Autoinflammatory bone disorders. Clinical Immunology. 2013;147(3):185-196. doi:10.1016/j.clim.2012.12.012
  • Tronconi E, Miniaci A, Baldazzi M, Greco L, Pession A. Biologic treatment for chronic recurrent multifocal osteomyelitis: report of four cases and review of the literature. Rheumatol Int. 2018;38(1):153-160. doi:10.1007/s00296-017-3877-0
  • Taddio A, Ferrara G, Insalaco A, et al. Dealing with Chronic Non-Bacterial Osteomyelitis: A practical approach. Pediatric Rheumatology. 2017;15(1). doi:10.1186/s12969-017-0216-7
  • De Vries E, Van Der Weij JP, Veen CJPVD, et al. In Vitro Effect of (3-Amino-l-Hydroxypropylidene)-1,1-Bisphosphomnc Acid (APD) on the Function of Mononuclear Phagocytes in Lymphocyte Proliferation.; 1982.
  • Santini D, Fratto ME, Vincenzi B, Cesa A La, Dianzani C, Tonini G. DRUG MECHANISMS AND TARGETS Bisphosphonate Effects in Cancer and Inflammatory Diseases In Vitro and In Vivo Modulation of Cytokine Activities. Vol 18.; 2004.
  • Girschick H, Finetti M, Orlando F, et al. The multifaceted presentation of chronic recurrent multifocal osteomyelitis: A series of 486 cases from the Eurofever international registry. Rheumatology (United Kingdom). 2018;57(7):1203-1211. doi:10.1093/rheumatology/key058
  • Sözeri B, Ayaz NA, Atıkan BY, et al. Clinical experiences in Turkish paediatric patients with chronic recurrent multifocal osteomyelitis. Turkish Journal of Pediatrics. 2019;61(6):879-884. doi:10.24953/turkjped.2019.06.009
  • Wipff J, Costantino F, Lemelle I, et al. A large national cohort of French patients with chronic recurrent multifocal osteitis. Arthritis and Rheumatology. 2015;67(4):1128-1137. doi:10.1002/art.39013
  • Sağ E, Sönmez HE, Demir S, et al. Chronic recurrent multifocal osteomyelitis in children: A single center experience over five years. Turkish Journal of Pediatrics. 2019;61(3):386-391. doi:10.24953/turkjped.2019.03.010
  • Khanna G, Sato TS, Ferguson P. Imaging of chronic recurrent multifocal osteomyelitis. Radiographics. 2009;29:1159-1177.
  • Hassan M, Assi H, Hassan M, et al. Chronic Recurrent Multifocal Osteomyelitis: A Comprehensive Literature Review. Cureus. Published online August 8, 2023. doi:10.7759/cureus.43118
  • Ulu K, İşgüder R, Karadağ ŞG, et al. Clinical characteristics and predictors for recurrence in chronic nonbacterial osteomyelitis: a retrospective multicenter analysis. Turk J Med Sci. 2023;53(5):1105-1111. doi:10.55730/1300-0144.5675
  • Zhao Y, Wu EY, Oliver MS, et al. Consensus Treatment Plans for Chronic Nonbacterial Osteomyelitis Refractory to Nonsteroidal Antiinflammatory Drugs and/or With Active Spinal Lesions. Arthritis Care Res (Hoboken). 2018;70(8):1228-1237. doi:10.1002/acr.23462
Toplam 19 adet kaynakça vardır.

Ayrıntılar

Birincil Dil Türkçe
Konular Klinik Tıp Bilimleri (Diğer)
Bölüm Araştırma Makaleleri
Yazarlar

Zeynep Özaslan 0000-0003-2641-4140

Nihal Şahin 0000-0002-2122-6952

Yonca Akgoz Anık 0000-0002-6768-2574

Hafize Emine Sönmez 0000-0002-9186-3068

Yayımlanma Tarihi 30 Haziran 2024
Gönderilme Tarihi 13 Kasım 2023
Kabul Tarihi 26 Nisan 2024
Yayımlandığı Sayı Yıl 2024 Cilt: 7 Sayı: 2

Kaynak Göster

AMA Özaslan Z, Şahin N, Anık YA, Sönmez HE. Çocuklarda Kronik Non Bakteriyel Osteomiyelit: Tek Merkez Deneyimi. Acta Med Nicomedia. Haziran 2024;7(2):169-174.

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