磁共振成像对初诊为幼年特发性关节炎的急性淋巴细胞白血病的诊断价值

doi:10.12372/jcp.2022.21e0851

摘要/Abstract

摘要：

目的探讨磁共振成像（MRI）对临床初诊为幼年特发性关节炎（JIA）的急性淋巴细胞白血病（ALL）患儿的诊断价值。方法回顾性分析2018年1月—2020年12月以下肢疼痛为首发症状且临床初诊为JIA,但最终经骨髓检查确诊为ALL患儿（病例组）的临床资料。将同时期确诊的JIA患儿作为对照组。所有患儿均在就诊初期（未进行相关治疗前）行双膝关节MRI扫描,探讨MRI对儿童ALL与JIA的鉴别诊断价值。结果病例组15例,男9例、女6例,中位年龄年龄5.1（3.6~7.8）岁,下肢关节疼痛病程25.0（16.5 ~35.5）天;对照组18例,男7例、女11例,中位年龄5.3（3.5~7.3）岁,下肢关节疼痛病程23.5（15.0~39.0）天。病例组中,13例患儿MRI可见双膝关节骨髓信号异常,多表现为弥漫性均匀或不均匀T1WI等低信号、T2WI高信号;2例MRI未见异常。对照组中,14例患儿MRI可见干骺端及骨骺信号异常,呈小斑片状T1WI等低信号、T2 WI高信号;4例MRI未见明显异常。以MRI特征中双膝关节病变弥漫分布为诊断标准,其诊断ALL的灵敏度和特异度为80.00%和94.44%;以T1WI等低信号及T2WI高信号为诊断标准,其灵敏度和特异度为86.67%和66.67%。病例组膝关节表观弥散系数（ADC）值低于对照组,差异有统计学意义（P<0.01）。ROC曲线分析显示,当ADC以0.88×10^-3 mm² /s为诊断阈值时,其诊断ALL的曲线下面积（AUC）为0.895,灵敏度为 86.7%,特异度为 83.3%,诊断准确率为 84.9%。结论 MRI作为一种无创的检查方法,对于以下肢疼痛为首发症状且实验室外周血检查无明显异常的ALL与JIA患儿的鉴别诊断具有较大临床价值。

关键词: 磁共振成像, 弥散加权成像, 急性淋巴细胞白血病, 幼年特发性关节炎, 下肢疼痛

Abstract:

Objective The aim of this study is to assess the diagnostic value of magnetic resonance imaging (MRI) in children with acute lymphoblastic leukemia (ALL) misdiagnosed as juvenile idiopathic arthritis (JIA). Methods Retrospective analysis was performed on children with lower extremity pains as the first symptom in our hospital from January 2018 to December 2020 who were initially diagnosed as JIA. Among them, fifteen patients were finally diagnosed as ALL confirmed by bone marrow aspiration, eighteen patients were JIA. All of them underwent blood routine and bilateral knee MRI examination before any treatment. The MRI were performed, including T1WI, spectral attenuated inversion recovery (SPAIR) T2WI and DWI (b value was 0 and 600 s/mm²). The MRI features of bone marrow were assessed by extent and location, the signal intensity, morphological features. The ADC value of all knee joints were measured and were compared between the two groups. The diagnostic value was calculated. Results There were no statistically significant differences in WBC count, hemoglobin (Hb), platelet count (PLT), C- creative protein (CRP) and erythrocyte sedimentation rate (ESR) between the case group and the control group (P>0.05). Abnormal signal intensity of bone marrow in bilateral bones of lower extremities was demonstrated in 13 patients with ALL patients which showed diffuse homogeneous iso- or low-signal intensity on T1W images and high signal intensity on T2 SPAIR images; other 2 patients had normal results of MRI. In 18 patients with JIA, 14 patients showed ill-defined patchy areas in the metaphysis and epiphysis with iso- or low-signal intensity on T1W images and high signal intensity on T2 SPAIR images; other 4 cases of JIA patients had normal results of MRI. The positive rate in the case group was higher than that in the control group. Compared with the control group, the MRI features of the case group were bilateral diffuse low signal intensity in T1W images and high signal intensity in T2W images (P<0.01). The ADC values in ALL group were lower than control group (t=-14.133, P<0.01). Taking 0.88×10^-3 mm² /s as the threshold, AUC of ROC curve in diagnosis of ALL was 0.895, with sensitivity, specificity and diagnostic accuracy was 86.7%, 83.3% and 84.9%. Conclusion MRI may serve as a noninvasive method to help discrimination of ALL from JIA in children presenting with lower extremity pains. MRI show diffuse abnormal signal intensity on bilateral lower extremities are more likely to be initial ALL.

Key words: MRI, DWI, acute lymphoblastic leukemia, juvenile idiopathic arthritis, lower extremities pain

徐琳, 孙爱敏, 黄华, 潘慧红, 钟玉敏. 磁共振成像对初诊为幼年特发性关节炎的急性淋巴细胞白血病的诊断价值[J]. 临床儿科杂志, 2022, 40(2): 107-112.

XU Lin, SUN Aimin, HUANG Hua, PAN Huihong, ZHONG Yumin. Diagnostic value of MRI in children with acute lymphoblastic leukemia misdiagnosed as juvenile idiopathic arthritis[J]. Journal of Clinical Pediatrics, 2022, 40(2): 107-112.

图/表 4

表1

图1

图2

表2

参考文献 22

[1]	Agodi A, Barchitta M, Trigilia C, et al. Neutrophil counts distinguish between malignancy and arthritis in children with musculoskeletal pain: a case-control study[J]. BMC Pediatr, 2013, 13:15. doi: 10.1186/1471-2431-13-15
[2]	Brix N, Rosthøj S, Herlin T, et al. Arthritis as presenting manifestation of acute lymphoblastic leukaemia in children[J]. Arch Dis Child, 2015, 100(9):821-825. doi: 10.1136/archdischild-2014-307751
[3]	Zombori L, Kovacs G, Csoka M, et al. Rheumatic symptoms in childhood leukaemia and lymphoma-a ten-year retrospective study[J]. Pediatr Rheumatology Online J, 2013; 11:20. doi: 10.1186/1546-0096-11-20
[4]	Kwee TC, Fijnheer R, Ludwig I, et al. Whole-body magnetic resonance imaging, including diffusion-weighted imaging, for diagnosing bone marrow involvement in malignant lymphoma[J]. Br J Haematol, 2010, 149(4):628-630. doi: 10.1111/bjh.2010.149.issue-4
[5]	Guillerman RP. Imaging of normal and abnormal bone marrow// Slovis TL, Adler BH, Bloom DA, et al, Eds. Cafey’s Pediatric Diagnostic Imaging[M]. Elsevier, Philadelphia, Pa, USA, 11th edition, 2008.
[6]	Yoshikawa T, Tanizawa A, Suzuki K, et al. The usefulness of T1- weighted magnetic resonance images for diagnosis of acute leukemia manifesting musculoskeletal symptoms prior to appearance of peripheral blood abnormalities[J]. Case Rep Pediatr, 2016: 2802596.
[7]	李琛, 曹兰芳, 薛海燕. 儿童急性淋巴细胞白血病和幼年特发性关节炎的初步鉴别[J]. 国际儿科学杂志, 2014(5):542-543.
[8]	李莎, 张伟. 急性淋巴细胞白血病误诊为幼年特发性关节炎并文献复习[J]. 国际输血及血液学杂志, 2017, 40(3):224-227.
[9]	Brix N, Hasle H, Rosthøj S, et al. Characteristics of children with acute lymphoblastic leukemia presenting with arthropathy[J]. Clin Rheumatol, 2018, 37(9):2455-2463. doi: 10.1007/s10067-018-4034-1
[10]	Tsujioka T, Sugiyama M, Ueki M, et al. Difficulty in the diagnosis of bone and joint pain associated with pediatric acute leukemia comparison with juvenile idiopathic arthritis[J]. Mod Rheumatol, 2018, 28(1):108-113. doi: 10.1080/14397595.2017.1332474 pmid: 28612674
[11]	Petty RE, Southwood TR, Manners P, et al. International league of associations for rheumatology classification of juvenileidiopathic arthritis: second revision, Edmonton, 2001[J]. J Rheumatol, 2004, 31(2):390-392. pmid: 14760812
[12]	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)[J]. Pediatr Rheumatol Online J, 2020, 18(1):1. doi: 10.1186/s12969-019-0376-8
[13]	Navarro SM, Matcuk GR, Patel DB, et al. Musculoskeletal imaging findings of hematologic malignancies[J]. Radiographics, 2017, 37(3):881-900. doi: 10.1148/rg.2017160133
[14]	Chan BY, Gill KG, Rebsamen SL, et al. MR Imaging of pediatric bone marrow[J]. Radiographics, 2016, 36(6):1911-1930. doi: 10.1148/rg.2016160056
[15]	Hemke R, Kuijpers TW, van den Berg JM, et al. The diagnostic accuracy of unenhanced MRI in the assessment of joint abnormalities in juvenile idiopathic arthritis[J]. Eur Radiol, 2013, 23(7):1998-2004. doi: 10.1007/s00330-013-2770-9
[16]	Attariwala R, Picker W. Whole body MRI: Improved lesion diction and characterization with diffusion weighted techniques[J]. J Magn Reson Imaging, 2013, 38(2):253-268. doi: 10.1002/jmri.24285 pmid: 23960006
[17]	Lee KH, Chung HY, Xu X, et al. Apparent diffusion coefficient as an imaging biomarker for spinal disease activity in axial spondyloarthritis[J]. Radiology, 2019, 291(1):121-128. doi: 10.1148/radiol.2019180960
[18]	Shi Z, Han J, Qin J, et al. Clinical application of diffusion-weighted imaging and dynamic contrast-enhanced MRI in assessing the clinical curative effect of early ankylosing spondylitis[J]. Medicine (Baltimore), 2019, 98(20):e15227. doi: 10.1097/MD.0000000000015227
[19]	Koutoulidis V, Fontara S, Terpos E, et al. Quantitative diffusion-weighted imaging of the bone marrow: an adjunct tool for the diagnosis of a diffuse MR imaging pattern in patients with multiple myeloma[J]. Radiology, 2017, 282(2):484-493. doi: 10.1148/radiol.2016160363 pmid: 27610934
[20]	Park S, Park JG, Jun S, et al. Differentiation of bone metastases from prostate cancer and benign red marrow depositions of the pelvic bone with multiparametric MRI[J]. Magn Reson Imaging, 2020, 73:118-124. doi: 10.1016/j.mri.2020.08.019
[21]	Nishii T, Kono AK, Akasaka Y, et al. Bone marrow magnetic resonance imaging of the clivus in pediatric leukemia patients and normal controls[J]. Jpn J Radiol, 2015, 233(3):146-152.
[22]	Cao W, Liang C, Gen Y, et al. Role of diffusion-weighted imaging for detecting bone marrow infiltration in skull in children with acute lymphoblastic leukemia[J]. Diagn Interv Radiol, 2016, 22(6):580-586. doi: 10.5152/dir

组别	例数	WBC/×10⁹·L^-1	Hb/g·L^-1	PLT/×10⁹·L^-1	CRP/mg·L^-1	ESR/mm·h^-1
病例组	15	6.4(4.9~13.6)	125.0(118.0~137.0)	284.0(195.0~300.0)	3.0(1.0~7.0)	5.0(2.0~30.0)
对照组	18	10.1(6.0~12.7)	121.5(111.0~137.0)	293.5(169.0~352.8)	2.5(1.0~38.9)	7.0(2.0~62.8)
Z值		1.09	0.63	0.77	1.27	1.84
P		0.442	0.656	0.513	0.929	0.066

组别	例数	T1WI				T2WI			病变分布
组别	例数	低信号	等低信号	等信号	正常	稍高信号	高信号	正常	双侧弥漫	双侧骨骺及干骺端	单侧骨骺及干骺端	正常
病例组	15	2(13.3)	11(73.3)	0(0)	2(13.3)	0(0)	13(86.7)	2(13.3)	12(80.0)	1(6.7)	0(0)	2(13.3)
对照组	18	0(0)	6(33.3)	8(44.4)	4(22.2)	8(44.4)	6(33.3)	4(22.2)	1(5.6)	7(38.9)	6(33.3)	4(22.2)
P¹⁾		0.003				0.001			<0.001