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Lack of bone lesions at diagnosis is associated with inferior outcome in multisystem langerhans cell histiocytosis of childhood

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Lack of bone lesions at diagnosis is associated with inferior outcome in multisystem langerhans cell histiocytosis of childhood. / Aricò, Maurizio; Astigarraga, Itziar; Braier, Jorge; Donadieu, Jean; Gadner, Helmut; Glogova, Evgenia; Grois, Nicole; Henter, Jan-Inge; Janka, Gritta; McClain, Kenneth L; Ladisch, Stephan; Pötschger, Ulrike; Rosso, Diego; Thiem, Elfriede; Weitzman, Sheila; Windebank, Kevin; Minkov, Milen; Histiocyte Society.

in: BRIT J HAEMATOL, Jahrgang 169, Nr. 2, 04.2015, S. 241-8.

Publikationen: SCORING: Beitrag in Fachzeitschrift/Zeitung › SCORING: Zeitschriftenaufsatz › Forschung › Begutachtung

Harvard

Aricò, M, Astigarraga, I, Braier, J, Donadieu, J, Gadner, H, Glogova, E, Grois, N, Henter, J-I, Janka, G, McClain, KL, Ladisch, S, Pötschger, U, Rosso, D, Thiem, E, Weitzman, S, Windebank, K, Minkov, M & Histiocyte Society 2015, 'Lack of bone lesions at diagnosis is associated with inferior outcome in multisystem langerhans cell histiocytosis of childhood', BRIT J HAEMATOL, Jg. 169, Nr. 2, S. 241-8. https://doi.org/10.1111/bjh.13271

APA

Aricò, M., Astigarraga, I., Braier, J., Donadieu, J., Gadner, H., Glogova, E., Grois, N., Henter, J-I., Janka, G., McClain, K. L., Ladisch, S., Pötschger, U., Rosso, D., Thiem, E., Weitzman, S., Windebank, K., Minkov, M., & Histiocyte Society (2015). Lack of bone lesions at diagnosis is associated with inferior outcome in multisystem langerhans cell histiocytosis of childhood. BRIT J HAEMATOL, 169(2), 241-8. https://doi.org/10.1111/bjh.13271

Vancouver

Aricò M, Astigarraga I, Braier J, Donadieu J, Gadner H, Glogova E et al. Lack of bone lesions at diagnosis is associated with inferior outcome in multisystem langerhans cell histiocytosis of childhood. BRIT J HAEMATOL. 2015 Apr;169(2):241-8. https://doi.org/10.1111/bjh.13271

Bibtex

@article{16317320d1224ecbafb1f2a5a7d24bac,
title = "Lack of bone lesions at diagnosis is associated with inferior outcome in multisystem langerhans cell histiocytosis of childhood",
abstract = "Skeletal involvement is generally, but not universally, characteristic of Langerhans cell histiocytosis (LCH). We investigated whether the presence of bone lesions at diagnosis is a prognostic factor for survival in LCH. Nine hundred and thirty-eight children with multisystem (MS) LCH, both high (386 RO+) and low (RO-) risk, were evaluated for bone lesions at diagnosis. Risk organ (RO+) involvement was defined as: haematopoietic system (haemoglobin <100 g/l, and/or white blood cell count <4·0 × 10(9) /l and/or platelet count <100 × 10(9) /l), spleen (>2 cm below the costal margin), liver (>3 cm and/or hypoproteinaemia, hypoalbuminaemia, hyperbilirubinaemia, and/or increased aspartate transaminase/alanine transaminase). Given the general view that prognosis in LCH worsens with increasing extent of disease, the surprising finding was that in MS+RO+ LCH the probability of survival with bone involvement 74 ± 3% (n = 230, 56 events) was reduced to 62 ± 4% (n = 156, 55 events) if this was absent (P = 0·007). An even greater difference was seen in the subgroup of patients with both liver and either haematopoiesis or spleen involvement: 61 ± 5% survival (n = 105; 52 events) if patients had bony lesions, versus 47 ± 5% (n = 111; 39 events) if they did not (P = 0·014). This difference was retained in multivariate analysis (P = 0·048). Although as yet unexplained, we conclude that bone involvement at diagnosis is a previously unrecognized favourable prognostic factor in MS+RO+ LCH.",
keywords = "Bone and Bones, Child, Child, Preschool, Histiocytosis, Langerhans-Cell, Humans, Infant, Prognosis, Proportional Hazards Models, Histiocytosis",
author = "Maurizio Aric{\`o} and Itziar Astigarraga and Jorge Braier and Jean Donadieu and Helmut Gadner and Evgenia Glogova and Nicole Grois and Jan-Inge Henter and Gritta Janka and McClain, {Kenneth L} and Stephan Ladisch and Ulrike P{\"o}tschger and Diego Rosso and Elfriede Thiem and Sheila Weitzman and Kevin Windebank and Milen Minkov and {Histiocyte Society}",
note = "{\textcopyright} 2014 John Wiley & Sons Ltd.",
year = "2015",
month = apr,
doi = "10.1111/bjh.13271",
language = "English",
volume = "169",
pages = "241--8",
journal = "BRIT J HAEMATOL",
issn = "0007-1048",
publisher = "Wiley-Blackwell",
number = "2",

}

RIS

TY - JOUR

T1 - Lack of bone lesions at diagnosis is associated with inferior outcome in multisystem langerhans cell histiocytosis of childhood

AU - Aricò, Maurizio

AU - Astigarraga, Itziar

AU - Braier, Jorge

AU - Donadieu, Jean

AU - Gadner, Helmut

AU - Glogova, Evgenia

AU - Grois, Nicole

AU - Henter, Jan-Inge

AU - Janka, Gritta

AU - McClain, Kenneth L

AU - Ladisch, Stephan

AU - Pötschger, Ulrike

AU - Rosso, Diego

AU - Thiem, Elfriede

AU - Weitzman, Sheila

AU - Windebank, Kevin

AU - Minkov, Milen

AU - Histiocyte Society

N1 - © 2014 John Wiley & Sons Ltd.

PY - 2015/4

Y1 - 2015/4

N2 - Skeletal involvement is generally, but not universally, characteristic of Langerhans cell histiocytosis (LCH). We investigated whether the presence of bone lesions at diagnosis is a prognostic factor for survival in LCH. Nine hundred and thirty-eight children with multisystem (MS) LCH, both high (386 RO+) and low (RO-) risk, were evaluated for bone lesions at diagnosis. Risk organ (RO+) involvement was defined as: haematopoietic system (haemoglobin <100 g/l, and/or white blood cell count <4·0 × 10(9) /l and/or platelet count <100 × 10(9) /l), spleen (>2 cm below the costal margin), liver (>3 cm and/or hypoproteinaemia, hypoalbuminaemia, hyperbilirubinaemia, and/or increased aspartate transaminase/alanine transaminase). Given the general view that prognosis in LCH worsens with increasing extent of disease, the surprising finding was that in MS+RO+ LCH the probability of survival with bone involvement 74 ± 3% (n = 230, 56 events) was reduced to 62 ± 4% (n = 156, 55 events) if this was absent (P = 0·007). An even greater difference was seen in the subgroup of patients with both liver and either haematopoiesis or spleen involvement: 61 ± 5% survival (n = 105; 52 events) if patients had bony lesions, versus 47 ± 5% (n = 111; 39 events) if they did not (P = 0·014). This difference was retained in multivariate analysis (P = 0·048). Although as yet unexplained, we conclude that bone involvement at diagnosis is a previously unrecognized favourable prognostic factor in MS+RO+ LCH.

AB - Skeletal involvement is generally, but not universally, characteristic of Langerhans cell histiocytosis (LCH). We investigated whether the presence of bone lesions at diagnosis is a prognostic factor for survival in LCH. Nine hundred and thirty-eight children with multisystem (MS) LCH, both high (386 RO+) and low (RO-) risk, were evaluated for bone lesions at diagnosis. Risk organ (RO+) involvement was defined as: haematopoietic system (haemoglobin <100 g/l, and/or white blood cell count <4·0 × 10(9) /l and/or platelet count <100 × 10(9) /l), spleen (>2 cm below the costal margin), liver (>3 cm and/or hypoproteinaemia, hypoalbuminaemia, hyperbilirubinaemia, and/or increased aspartate transaminase/alanine transaminase). Given the general view that prognosis in LCH worsens with increasing extent of disease, the surprising finding was that in MS+RO+ LCH the probability of survival with bone involvement 74 ± 3% (n = 230, 56 events) was reduced to 62 ± 4% (n = 156, 55 events) if this was absent (P = 0·007). An even greater difference was seen in the subgroup of patients with both liver and either haematopoiesis or spleen involvement: 61 ± 5% survival (n = 105; 52 events) if patients had bony lesions, versus 47 ± 5% (n = 111; 39 events) if they did not (P = 0·014). This difference was retained in multivariate analysis (P = 0·048). Although as yet unexplained, we conclude that bone involvement at diagnosis is a previously unrecognized favourable prognostic factor in MS+RO+ LCH.

KW - Bone and Bones

KW - Child

KW - Child, Preschool

KW - Histiocytosis, Langerhans-Cell

KW - Humans

KW - Infant

KW - Prognosis

KW - Proportional Hazards Models

KW - Histiocytosis

U2 - 10.1111/bjh.13271

DO - 10.1111/bjh.13271

M3 - SCORING: Journal article

C2 - 25522229

VL - 169

SP - 241

EP - 248

JO - BRIT J HAEMATOL

JF - BRIT J HAEMATOL

SN - 0007-1048

IS - 2

ER -