Chronic recurrent multifocal osteomyelitis in children with hypophosphatasia explained by anti-inflammatory nucleotidase activity of tissue nonspecific alkaline phosphatase in mesenchymal and hematopoietic cells

2019 ◽  
Author(s):  
Laurence Bessueille ◽  
Anne Briolay ◽  
Juna Como ◽  
Cylia Mansouri ◽  
Marie Gleizes ◽  
...  
Keyword(s):  
Alkaline Phosphatase ◽  
Hematopoietic Cells ◽  
Chronic Recurrent Multifocal Osteomyelitis ◽  
Nucleotidase Activity ◽  
Multifocal Osteomyelitis ◽  
Tissue Nonspecific Alkaline Phosphatase ◽  
Anti Inflammatory

scholarly journals Chronic Recurrent Multifocal Osteomyelitis of the First Metatarsal Bone: A Case Report

2009 ◽  
Vol 17 (1) ◽  
pp. 119-122 ◽  
Author(s):  
CK Chiu ◽  
VA Singh
Keyword(s):  
Case Report ◽  
Symptomatic Relief ◽  
Chronic Recurrent Multifocal Osteomyelitis ◽  
Metatarsal Bone ◽  
First Metatarsal ◽  
History Of ◽  
Multifocal Osteomyelitis ◽  
Inflammatory Drugs ◽  
Anti Inflammatory

We report a case of chronic recurrent multifocal osteomyelitis in a 9-year-old girl. She presented with a 9-month history of gradually worsening pain and swelling in her left foot. Non-steroidal anti-inflammatory drugs were prescribed but the symptoms persisted. She underwent curettage through a small oval corticotomy window on the first metatarsal bone. The pain and swelling improved promptly and she was able to walk without pain 2 weeks later. Curettage enabled rapid symptomatic relief and induced remission, with little risk of complications.

scholarly journals Interactive roles of CD73 and tissue nonspecific alkaline phosphatase in the renal vascular metabolism of 5′-AMP

2014 ◽  
Vol 307 (6) ◽  
pp. F680-F685 ◽  
Author(s):  
Edwin K. Jackson ◽  
Dongmei Cheng ◽  
Jonathan D. Verrier ◽  
Keri Janesko-Feldman ◽  
Patrick M. Kochanek
Keyword(s):  
Mass Spectrometry ◽  
Alkaline Phosphatase ◽  
Brain Tissue ◽  
Nucleotidase Activity ◽  
Wild Type ◽  
Renal Vasculature ◽  
Per Se ◽  
Tissue Nonspecific Alkaline Phosphatase ◽  
Renal Vascular

CD73 metabolizes extracellular 5′-AMP to adenosine; yet recent experiments in brain tissue suggest that CD73 is not required for the metabolism of 5′-AMP to adenosine because of tissue nonspecific alkaline phosphatase (TNAP), which like CD73 is a GPI-anchored ecto-enyzme with 5′-nucleotidase activity. Because adenosine importantly regulates renovascular function, we investigated whether both TNAP and CD73 are involved in the renovascular metabolism of 5′-AMP. To test this, we examined in isolated, perfused mouse kidneys the metabolism of 5′-AMP (applied to the lumen of the renal vasculature via intrarenal artery administration) to adenosine by measuring renal venous levels of 5′-AMP, adenosine, and inosine (adenosine metabolite) by mass spectrometry. In one study, we compared 5′-AMP metabolism in naive CD73+/+ (wild-type, n = 16) vs. CD73−/− (knockout, n = 16) kidneys; and in a second study, we compared 5′-AMP metabolism in CD73+/+ ( n = 9) vs. CD73−/− ( n = 8) kidneys pretreated with levamisole (1 mmol/l; TNAP inhibitor). In naive kidneys, 5′-AMP increased renal venous 5′-AMP, adenosine, and inosine, and these responses were similar in CD73+/+ vs. CD73−/− kidneys. Levamisole per se did not inhibit renovascular 5′-AMP metabolism; however, in the presence of levamisole, 5′-AMP increased renal venous 5′-AMP threefold more in CD73−/− vs. CD73+/+ kidneys and knockout of CD73 inhibited 5′-induced adenosine and inosine by 81 and 86%, respectively. TNAP mRNA, protein, and activity were similar in CD73+/+ vs. CD73−/− kidneys. In conclusion, CD73 and TNAP play interactive roles to metabolize luminally applied 5′-AMP in the renal vasculature such that inhibition of both is required to inhibit the production of adenosine.

scholarly journals POS1306 TREATMENT STRATEGIES IN CHRONIC RECURRENT MULTIFOCAL OSTEOMYELITIS (CMRO) OR CHRONIC NON-BACTERIAL OSTEOMYELITIS (CNO): SYSTEMATIC REVIEW AND META-ANALYSIS

2021 ◽  
Vol 80 (Suppl 1) ◽  
pp. 935.1-935
Author(s):  
R. Dos-Santos ◽  
C. Gomez-Vieites ◽  
D. Fernández Fernández ◽  
I. González Fernández ◽  
A. Souto Vilas ◽  
...  
Keyword(s):  
Treatment Guidelines ◽  
Meta Analysis ◽  
Chronic Recurrent Multifocal Osteomyelitis ◽  
Response Rates ◽  
Complete Response ◽  
Treatment Recommendation ◽  
Remission Rates ◽  
Multifocal Osteomyelitis ◽  
Inflammatory Drugs ◽  
Anti Inflammatory

Background:Glucocorticoids (GC), bisphosphonates (BP), non-steroidal anti-inflammatory drugs (NSAID) and classical synthetic or biological disease-modifying antirheumatic drugs (cs/bDMARD) have been employed in the treatment of chronic recurrent multifocal osteomyelitis (CRMO) or chronic non-bacterial osteomyelitis (CNO).1, 2 This one is a rare bone childhood illness and none treatment guidelines have been carried out till present.3Objectives:To asses which treatment schedule employed in CRMO had the best response rates and try to expose a treatment recommendation.Methods:A systematic literature review was made using Medline, Embase, Cochrane library and the Web of Science databases. The search strategy focused on synonyms of CRMO. A prevalence meta-analysis was performed to evaluate each treatment response. Stata 15.1 was used to perform statistical analysis.Results:The search identified 1883 articles, of which 43 were finally selected. Complete response rate reached with NSAIDs was acceptable [50% (CI95% 40-60)]. Lower response rates were reached by GC treatment [44% (CI95% 25-63)] or csDMARD [38% (CI95% 28-48)]. The best complete response rates were reached by bDMARD and BP treatments [69% (CI95% 56-82) and 73% (CI95% 62-84), respectively].Conclusion:This review and meta-analysis supports, taking into account its remission rates and its risk-benefit profile, NSAIDs as potential first-line agents in CRMO treatment. bDMARD and BP have reached the higher remission rates, turning into helpful treatment alternatives. There is not any treatment guidelines driving CRMO patients, but this analysis could help to select a suitable agent for each patient. Decision-making should be individualized.References:[1]Wipff J, Costantino F, Lemelle I, et al. A large national cohort of French patients with chronic recurrent multifocal osteitis. Arthritis & Rheumatol. 2014;67(4):1128-1137.[2]Zhao Y, Wu E Y, Oliver M S, et al. Consensus treatment plans for chronic nonbacterial osteomielitis refractory to nonsteroideal anti-inflammatory drugs and/or with active spinal lesions. Arthritis Care Res. 2018;70(8):1228-1237.[3]Menashe SJ, Aboughalia H, Zhao Y, et al. The many faces of pediatric chronic recurrent multifocal osteomyelitis (CRMO): a practical location- and case-based approach to differenciate CRMO from its mimics. J Magn Reson Imaging. 2020;e27299.Disclosure of Interests:None declared

scholarly journals Tissue-nonspecific alkaline phosphatase is an anti-inflammatory nucleotidase

Bone ◽  
2020 ◽  
Vol 133 ◽  
pp. 115262 ◽  
Author(s):  
L. Bessueille ◽  
A. Briolay ◽  
J. Como ◽  
S. Mebarek ◽  
C. Mansouri ◽  
...  
Keyword(s):  
Alkaline Phosphatase ◽  
Tissue Nonspecific Alkaline Phosphatase ◽  
Anti Inflammatory

Chronic recurrent multifocal osteomyelitis in children: a new, MRI-based method of quantifying inflammation in the bone

2015 ◽  
Author(s):  
Dionysios Alexandrou ◽  
Benjamin Jacobs ◽  
O'Donnell Paul ◽  
Rikin Hargunani ◽  
Ananya Guha ◽  
...  
Keyword(s):  
Chronic Recurrent Multifocal Osteomyelitis ◽  
Multifocal Osteomyelitis

Chronic recurrent multifocal osteomyelitis in children

1988 ◽  
Vol 29 (5) ◽  
pp. 565-570 ◽  
Author(s):  
W. Mortensson ◽  
G. Edeburn ◽  
M. Fries ◽  
R. Nilsson
Keyword(s):  
Chronic Recurrent Multifocal Osteomyelitis ◽  
Multifocal Osteomyelitis

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

2021 ◽  
Author(s):  
Bartłomiej Juszczak ◽  
Jerzy Sułko
Keyword(s):  
Adverse Drug Reactions ◽  
Social Life ◽  
Chronic Recurrent Multifocal Osteomyelitis ◽  
Daily Activities ◽  
Drug Reactions ◽  
Patient Reported ◽  
Multifocal Osteomyelitis ◽  
Drug Dependent ◽  
Treatment Onset ◽  
The Impact

AbstractTo evaluate patient-reported effectiveness, safety and social influence of Pamidronate in the therapy of NSAIDs-refractory Chronic Recurrent Multifocal Osteomyelitis in children. Authors reviewed self-created questionnaires, which asked patients for symptoms alleviation, adverse drug reactions frequency and degree of severity and daily activities self-reliance. Only surveys with complete answers, which were returned to authors by an e-mail from juvenile patients treated for NSAIDs-refractory Chronic Recurrent Multifocal Osteomyelitis at the University Children’s Hospital of Cracow were analyzed. Between 2010 and 2019, 61 children were diagnosed with NSAIDs-refractory Chronic Recurrent Multifocal Osteomyelitis at our department. Out of 61 requests sent, 42 complete replies (33 females, 9 males) were gathered and analyzed. All patients included in this research were administered with at least one set of Pamidronate intravenously in the dose of 1 mg/kg/day for 3 consecutive days. Our analysis shows remarkable in terms of patient’s impressions decrease of pain intensity after 2.5 series of Pamidronate on average, and total pain resolution after 5.9 series on average. Overall number of adverse drug reaction events reported by responders was 105. One patient developed drug-dependent renal insufficiency in the course of therapy. Outcome assessment indicates that nearly 50% of the studied population was more eager to participate in social life just after the first infusion of the drug. 95% of the surveyed unanimously agreed to recommend Pamidronate therapy to cure NSAIDs-refractory CRMO. 39 out of 42 (93%) patients considered Pamidronate effective at the end of the treatment. Onset of Pamidronate’s action is gradual and differs in terms of symptoms alleviation between sexes. The therapy can induce considerable number of adverse drug reactions (2.5 per patient). Only 3 out of 42 (7%) patients were free from any ADRs. To demonstrate the impact of the use of Pamidronate on daily activities more precisely, further research with quantification of the quality of life is warranted.

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