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Persistent osteoarticular pain in children: early clinical and laboratory findings suggestive of acute lymphoblastic leukemia (a multicenter case-control study of 147 patients).
Pediatr Rheumatol Online J. 2020 Jan 02; 18(1):1.PR

Abstract

BACKGROUND

The aim of this study was to identify early clinical and laboratory features that distinguish acute lymphoblastic leukemia (ALL) from juvenile idiopathic arthritis (JIA) in children presenting with persistent bone or joint pain for at least 1 month.

METHODS

We performed a multicenter case-control study and reviewed medical records of children who initially presented with bone or joint pain lasting for at least 1 month, all of whom were given a secondary diagnosis of JIA or ALL, in four French University Hospitals. Each patient with ALL was paired by age with two children with JIA. Logistic regression was used to compare clinical and laboratory data from the two groups.

RESULTS

Forty-nine children with ALL and 98 with JIA were included. The single most important feature distinguishing ALL from JIA was the presence of hepatomegaly, splenomegaly or lymphadenopathy; at least one of these manifestations was present in 37 cases with ALL, but only in 2 controls with JIA, for an odds ratio (OR) of 154 [95%CI: 30-793] (regression coefficient: 5.0). If the presence of these findings is missed or disregarded, multivariate analyses showed that non-articular bone pain and/or general symptoms (asthenia, anorexia or weight loss) (regression coefficient: 4.8, OR 124 [95%CI: 11.4-236]), neutrophils < 2 × 109/L (regression coefficient: 3.9, OR 50 [95%CI: 4.3-58]), and platelets < 300 × 109/L (regression coefficient: 2.6, OR 14 [95%CI: 2.3-83.9]) were associated with the presence of ALL (area under the ROC curve: 0.96 [95%CI: 0.93-0.99]).

CONCLUSIONS

Based on our findings we propose the following preliminary decision tree to be tested in prospective studies: in children presenting with at least 1 month of osteoarticular pain and no obvious ALL in peripheral smear, perform a bone marrow examination if hepatomegaly, splenomegaly or lymphadenopathy is present. If these manifestations are absent, perform a bone marrow examination if there is fever or elevated inflammatory markers associated with non-articular bone pain, general symptoms (asthenia, anorexia or weight loss), neutrophils < 2 × 109/L or platelets < 300 × 109/L.

Authors+Show Affiliations

Unité d'Onco-Hémato-Immunologie pédiatrique, CHU Angers, 4 rue Larrey, 49933, Angers, France. louvignemathilde@outlook.fr.Unité d'Onco-Hémato-Immunologie pédiatrique, CHU Angers, 4 rue Larrey, 49933, Angers, France.Service de Pédiatrie générale, CHU Angers, Angers, France.Unité d'Onco-Hémato-Immunologie pédiatrique, CHU Rennes, Rennes, France.Unité d'Onco-Hémato-Immunologie pédiatrique, CHU Angers, 4 rue Larrey, 49933, Angers, France.Unité d'Onco-Hémato-Immunologie pédiatrique, CHU Nantes, Nantes, France.Unité d'Hémato-Immunologie pédiatrique, CHU Robert Debré, Hôpitaux de Paris, Paris, France.Unité d'Hémato-Immunologie pédiatrique, CHU Robert Debré, Hôpitaux de Paris, Paris, France. Université de Paris, UFR Paris Diderot, Paris, France.Service de Pédiatrie générale, CHU Rennes, Rennes, France.Service de Pédiatrie générale, CHU Rennes, Rennes, France.Service de Pédiatrie générale, CHU Rennes, Rennes, France.Université de Paris, UFR Paris Diderot, Paris, France. Service de Pédiatrie générale Maladies Infectieuses et Médecine Interne, Centre de référence des rhumatismes inflammatoires et maladies auto-immunes systémiques rares de l'enfant RAISE, CHU Robert Debré, Hôpitaux de Paris, Paris, France.Service de Pédiatrie générale Maladies Infectieuses et Médecine Interne, Centre de référence des rhumatismes inflammatoires et maladies auto-immunes systémiques rares de l'enfant RAISE, CHU Robert Debré, Hôpitaux de Paris, Paris, France.Service de Pédiatrie générale Maladies Infectieuses et Médecine Interne, Centre de référence des rhumatismes inflammatoires et maladies auto-immunes systémiques rares de l'enfant RAISE, CHU Robert Debré, Hôpitaux de Paris, Paris, France.Service de Pédiatrie générale Maladies Infectieuses et Médecine Interne, Centre de référence des rhumatismes inflammatoires et maladies auto-immunes systémiques rares de l'enfant RAISE, CHU Robert Debré, Hôpitaux de Paris, Paris, France.Unité d'Immuno-Hématologie et Rhumatologie Pédiatriques, Hôpital Necker-Enfants Malades, Hôpitaux de Paris, Paris, France.Unité d'Onco-Hémato-Immunologie pédiatrique, CHU Rennes, Rennes, France.Unité d'Onco-Hémato-Immunologie pédiatrique, CHU Angers, 4 rue Larrey, 49933, Angers, France.No affiliation info available

Pub Type(s)

Journal Article
Multicenter Study

Language

eng

PubMed ID

31898528

Citation

Louvigné, Mathilde, et al. "Persistent Osteoarticular Pain in Children: Early Clinical and Laboratory Findings Suggestive of Acute Lymphoblastic Leukemia (a Multicenter Case-control Study of 147 Patients)." Pediatric Rheumatology Online Journal, vol. 18, no. 1, 2020, p. 1.
Louvigné M, Rakotonjanahary J, Goumy L, et al. Persistent osteoarticular pain in children: early clinical and laboratory findings suggestive of acute lymphoblastic leukemia (a multicenter case-control study of 147 patients). Pediatr Rheumatol Online J. 2020;18(1):1.
Louvigné, M., Rakotonjanahary, J., Goumy, L., Tavenard, A., Brasme, J. F., Rialland, F., Baruchel, A., Auclerc, M. F., Despert, V., Desgranges, M., Jean, S., Faye, A., Meinzer, U., Lorrot, M., Job-Deslandre, C., Bader-Meunier, B., Gandemer, V., & Pellier, I. (2020). Persistent osteoarticular pain in children: early clinical and laboratory findings suggestive of acute lymphoblastic leukemia (a multicenter case-control study of 147 patients). Pediatric Rheumatology Online Journal, 18(1), 1. https://doi.org/10.1186/s12969-019-0376-8
Louvigné M, et al. Persistent Osteoarticular Pain in Children: Early Clinical and Laboratory Findings Suggestive of Acute Lymphoblastic Leukemia (a Multicenter Case-control Study of 147 Patients). Pediatr Rheumatol Online J. 2020 Jan 2;18(1):1. PubMed PMID: 31898528.
* Article titles in AMA citation format should be in sentence-case
TY - JOUR T1 - Persistent osteoarticular pain in children: early clinical and laboratory findings suggestive of acute lymphoblastic leukemia (a multicenter case-control study of 147 patients). AU - Louvigné,Mathilde, AU - Rakotonjanahary,Josué, AU - Goumy,Laurence, AU - Tavenard,Aude, AU - Brasme,Jean-François, AU - Rialland,Fanny, AU - Baruchel,André, AU - Auclerc,Marie-Françoise, AU - Despert,Véronique, AU - Desgranges,Marie, AU - Jean,Sylvie, AU - Faye,Albert, AU - Meinzer,Ulrich, AU - Lorrot,Mathie, AU - Job-Deslandre,Chantal, AU - Bader-Meunier,Brigitte, AU - Gandemer,Virginie, AU - Pellier,Isabelle, AU - ,, Y1 - 2020/01/02/ PY - 2019/07/09/received PY - 2019/10/29/accepted PY - 2020/1/4/entrez PY - 2020/1/4/pubmed PY - 2020/11/20/medline KW - Arthralgia KW - Bone pain KW - Childhood acute lymphoblastic leukemia KW - Diagnosis KW - Juvenile idiopathic arthritis SP - 1 EP - 1 JF - Pediatric rheumatology online journal JO - Pediatr Rheumatol Online J VL - 18 IS - 1 N2 - BACKGROUND: The aim of this study was to identify early clinical and laboratory features that distinguish acute lymphoblastic leukemia (ALL) from juvenile idiopathic arthritis (JIA) in children presenting with persistent bone or joint pain for at least 1 month. METHODS: We performed a multicenter case-control study and reviewed medical records of children who initially presented with bone or joint pain lasting for at least 1 month, all of whom were given a secondary diagnosis of JIA or ALL, in four French University Hospitals. Each patient with ALL was paired by age with two children with JIA. Logistic regression was used to compare clinical and laboratory data from the two groups. RESULTS: Forty-nine children with ALL and 98 with JIA were included. The single most important feature distinguishing ALL from JIA was the presence of hepatomegaly, splenomegaly or lymphadenopathy; at least one of these manifestations was present in 37 cases with ALL, but only in 2 controls with JIA, for an odds ratio (OR) of 154 [95%CI: 30-793] (regression coefficient: 5.0). If the presence of these findings is missed or disregarded, multivariate analyses showed that non-articular bone pain and/or general symptoms (asthenia, anorexia or weight loss) (regression coefficient: 4.8, OR 124 [95%CI: 11.4-236]), neutrophils < 2 × 109/L (regression coefficient: 3.9, OR 50 [95%CI: 4.3-58]), and platelets < 300 × 109/L (regression coefficient: 2.6, OR 14 [95%CI: 2.3-83.9]) were associated with the presence of ALL (area under the ROC curve: 0.96 [95%CI: 0.93-0.99]). CONCLUSIONS: Based on our findings we propose the following preliminary decision tree to be tested in prospective studies: in children presenting with at least 1 month of osteoarticular pain and no obvious ALL in peripheral smear, perform a bone marrow examination if hepatomegaly, splenomegaly or lymphadenopathy is present. If these manifestations are absent, perform a bone marrow examination if there is fever or elevated inflammatory markers associated with non-articular bone pain, general symptoms (asthenia, anorexia or weight loss), neutrophils < 2 × 109/L or platelets < 300 × 109/L. SN - 1546-0096 UR - https://www.unboundmedicine.com/medline/citation/31898528/Persistent_osteoarticular_pain_in_children:_early_clinical_and_laboratory_findings_suggestive_of_acute_lymphoblastic_leukemia__a_multicenter_case_control_study_of_147_patients__ L2 - https://ped-rheum.biomedcentral.com/articles/10.1186/s12969-019-0376-8 DB - PRIME DP - Unbound Medicine ER -