Eleven cases of dermatomyositis accompanied with gynecological malignant tumor: clinical characteristics and short-term follow-up

doi:10.16150/j.1671-2870.2021.01.008

Abstract

Abstract:

Objective: To analyze the clinical characteristics, diagnosis and treatment of dermatomyositis (DM) accompanied with gynecological malignant tumor to provide evidence for early detection and intervention of the disease. Methods: The clinical data of 11 DM patients with gynecological malignant tumor treated in our hospital from 2015 to 2020 were enrolled, and the clinical characteristics and treatment protocols were analyzed. Results: In eleven patients diagnosed DM accompanied with gynecological malignant tumor, three types of cancer were identified, including endometrial cancer (4/11), cervical cancer (2/11), and ovarian cancer (5/11). Eightout of 11 patients were diagnosed stage Ⅲ-Ⅳ gynecological cancer according to 2020 FIGO(International Federation of Gynecology and Obstetrics) gynecological tumor staging. Ten patients were screened for gynecological cancer by imaging and tumor biomarkers,and diagnosed gynecological cancer after being confirmed as DM 1 week to 21 months. The other one patient was found to have ovarian cancer at 21^st month after diagnosis of DM because of the increased level of CA12-5 (carbohydrate antigen12-5) during the follow-up, however, her CT imaging of the pelvic and abdominal cavity and tumor biomarkers were normal in initial screening. The anti-TIF1-γ antibodies in 8 DM patients were tested, and 6 of them were positive. Except one patient with cervical cancer receiving directly radical radiotherapy and chemotherapy, the other 10 patients received gynecological tumor resection. The overall survival rate was 100% during the follow-up of 2 months to 2 years. There was no tumor recurrence duringthe follow-up, but the DM recurrence appeared in 2patients. Conclusions: For the DM patients lacking signs of malignant tumor in the initial screening, the continuing follow-upand screening for tumor biomarkers should get more attention.The DM patients accompanied gynecological malignant tumor often showed late-stage cancer (Ⅲ-Ⅳ staging) and the treatment should be mainly focused on the gynecological tumor.

Key words: Dermatomyositis, Gynecological malignancy, Cancer antigen12-5

CLC Number:

R737.31

LIU Chang, CAO Hua, FENG Weiwei, SHEN Lifei. Eleven cases of dermatomyositis accompanied with gynecological malignant tumor: clinical characteristics and short-term follow-up[J]. Journal of Diagnostics Concepts & Practice, 2021, 20(01): 53-59.

Figures/Tables 5

序号	妇科肿瘤	肿瘤分期	术前治疗方案	围手术期治疗方案
1	卵巢癌	卵巢及输卵管高级别浆液性乳头状癌ⅢC期	强的松8片（每日1次）+羟氯喹2片（每日1次）+ 帕夫林2片（每日3次）+艾洛松外用	围手术期未改变激素剂量,术后复查,逐渐减量
2	卵巢癌	卵巢高级别浆液性癌 ⅢC期	甲强龙28 mg（每日1次）+羟氯喹2片（每日1次）+ 沙利度胺50 mg（每日1次）+帕夫林2片（每日1次）	术前得宝松1支肌肉注射,沙利度胺、帕夫林及羟氯喹不停,直到2020年4月DM复发
3	卵巢癌	卵巢及早输卵管高级别浆液性癌ⅡA期	强的松15 mg（每日1次）+阿法骨化醇软胶囊 0.25 μg（每日1次）+羟氯喹每日2次）+ 沙利度胺50 mg（每日1次）	术前得宝松1支肌注,维持激素强的松3片（每日1次）,继续沙利度胺50 mg（每晚1次）, 帕夫林2片（每日2次）及羟氯喹2片（每日 2次）,术后强的松15 mg（每日1次）
4	卵巢癌	卵巢及输卵管高级别浆液性癌ⅢC期	强的松3/4片（每日1次）+帕夫林2片（每日1次）+ 羟氯喹2片（每日1次）	强的松15 mg,术前1天至术后3天,每日强的松 6片或甲强龙24 mg静脉滴注;第2次手术术前肌肉注射得宝松1支,术后第4日恢复强的松
5	卵巢癌	卵巢高级别浆液性癌 ⅢC期	甲泼尼龙24 mg（每日1次）	强的松5片（每日1次）;手术当日10片（每日 1次）,第2天5片（每日1次）
6	宫颈癌	宫颈鳞癌ⅣA期	强的松3片（每日3次）	未手术
7	宫颈癌	宫颈鳞癌ⅠB2	强的松6片（每日1次）	术前甲强龙40 mg静脉滴注,手术当日80 mg静脉滴注,术后40 mg静脉滴注3 d;术后第4天起强的松6片（每日1次）,后减至3片（每日1次）
8	子宫内膜癌	子宫内膜浆液性癌ⅢC2	强的松20 mg（每日2次）+他克莫司1 mg （每日2次）	停用他克莫司,手术当天及术后第2天甲强龙 40 mg（每日1次）,第3天起20 mg,之后强的松5片（每日1次）
9	子宫内膜癌	子宫内膜样腺癌ⅢC2期	强的松6片（每日1次）	手术当日至术后3 d每日甲强龙40 mg静脉滴注,术后第4天恢复强的松6片（每日1次）
10	子宫内膜癌	子宫内膜样腺癌Ⅱ期, 部分透明细胞癌	羟氯喹1片（每日2次）+甲强龙30 mg（每日1次）	术前得宝松1支肌肉注射
11	子宫内膜癌	子宫内膜癌肉瘤ⅢA期	强的松8片（每日1次）+羟氯喹2片（每日1次） +帕夫林2片（每日2次）+沙利度胺50 mg（每日1次）	术前1天、手术当日至术后3 d,甲强龙40 mg; 术后口服强的松30 mg（每日1次）,化疗结束后20 mg（每日1次）

References 18

[1]	中华医学会风湿病学分会. 多发性肌炎和皮肌炎诊断及治疗指南[J]. 中华风湿病学杂志, 2010, 14(12):828-831.
[2]	Barnes BE, Mawr B. Dermatomyositis and malignancy. A review of the literature[J]. Ann Intern Med, 1976, 84(1):68-76. pmid: 1106291
[3]	Aussy A, Boyer O, Cordel N. Dermatomyositis and immune-mediated necrotizing myopathies: a window on autoimmunity and cancer[J]. Front Immunol, 2017, 8:992. doi: 10.3389/fimmu.2017.00992 URL
[4]	Pinal-Fernandez I, Ferrer-Fabregas B, Trallero-Araguas E, et al. Tumour TIF1 mutations and loss of heterozygosity related to cancer-associated myositis[J]. Rheumatology (Oxford), 2018, 57(2):388-396. doi: 10.1093/rheumatology/kex413 pmid: 29149307
[5]	Nikolaos T, Maria T, Ioannis KD, et al. Dermatomyositis as an early manifestation and a significant clinical precursor of lung cancer: report of a rare case and review of the current literature[J]. Int J Clin Exp Med, 2013, 6(2):105-109.
[6]	Dalmau J, Graus F, Rosenblum MK, et al. Anti-Hu--associated paraneoplastic encephalomyelitis/sensory neuronopathy. A clinical study of 71 patients[J]. Medicine (Baltimore), 1992, 71(2):59-72. doi: 10.1097/00005792-199203000-00001 URL
[7]	Peterson K, Rosenblum MK, Kotanides H, et al. Paraneoplastic cerebellar degeneration. I. A clinical analysis of 55 anti-Yo antibody-positive patients[J]. Neurology, 1992, 42(10):1931-1937. doi: 10.1212/wnl.42.10.1931 pmid: 1407575
[8]	Casciola-Rosen L, Nagaraju K, Plotz P, et al. Enhanced autoantigen expression in regenerating muscle cells in idiopathic inflammatory myopathy[J]. J Exp Med, 2005, 201(4):591-601. doi: 10.1084/jem.20041367 pmid: 15728237
[9]	Levine SM. Cancer and myositis: new insights into an old association[J]. Curr Opin Rheumatol, 2006, 18(6):620-624. doi: 10.1097/01.bor.0000245721.02512.77 URL
[10]	Kim Y, Song KS, Sohn EH, et al. Anti-TIF1γ antibody and the expression of TIF1γ in idiopathic inflammatory myopathies[J]. Int J Rheum Dis, 2019, 22(2):314-320. doi: 10.1111/1756-185X.13424 URL
[11]	Scholtissek B, Ferring-Schmitt S, Maier J, et al. Expression of the autoantigen TRIM33/TIF1γ in skin and muscle of patients with dermatomyositis is upregulated, together with markers of cellular stress[J]. Clin Exp Dermatol, 2017, 42(6):659-662. doi: 10.1111/ced.13180 pmid: 28639716
[12]	赵辨. 中国临床皮肤病学[M]. 南京: 江苏科学技术出版社, 2010:807-814.
[13]	de Souza FH, Shinjo SK. Newly diagnosed dermatomyositis in the elderly as predictor of malignancy[J]. Rev Bras Reumatol, 2012, 52(5):713-721.
[14]	Buchbinder R, Forbes A, Hall S, et al. Incidence of malignant disease in biopsy-proven inflammatory myopathy. A population-based cohort study[J]. Ann Intern Med, 2001, 134(12):1087-1095. pmid: 11412048
[15]	Hill CL, Zhang Y, Sigurgeirsson B, et al. Frequency of specific cancer types in dermatomyositis and polymyositis: a population-based study[J]. Lancet, 2001, 357(9250):96-100. pmid: 11197446
[16]	夏群力, 刁立诚, 吴海曦, 等. 抗转录中介因子1-γ抗体是成人皮肌炎合并恶性肿瘤的血清学标志物[J]. 诊断学理论与实践, 2020, 19(3):274-278.
[17]	赵培珠, 李玉叶. 多发性肌炎/皮肌炎伴发恶性肿瘤的治疗[J]. 中国临床医生杂志, 2016, 44(77):8-10.
[18]	钟宗良, 莫浩元, 麦海强, 等. 初治鼻咽癌合并皮肌炎86例临床配对研究[J]. 中国肿瘤, 2012, 21(3):220-224.

序号	妇科肿瘤	年龄（岁）	绝经	其他合并症	恶性肿瘤家族史	皮肤病家族史
1	卵巢癌	78	已	冠心病、青光眼	无	无
2	卵巢癌	48	未	乙型肝炎	三姨患食管癌	无
3	卵巢癌	56	已	无	无	无
4	卵巢癌	54	已	肺结核	父亲患胃癌,同母异父哥哥患胃癌	无
5	卵巢癌	62	已	无	无	无
6	宫颈癌	55	已	乙型肝炎	无	无
7	宫颈癌	51	未	无	无	无
8	子宫内膜癌	66	已	无	无	无
9	子宫内膜癌	46	未	胃溃疡、脂肪肝	无	无
10	子宫内膜癌	68	已	无	无	无
11	子宫内膜癌	52	已	无	母亲患白血病,父亲患食管肿瘤	无

序号	妇科肿瘤	DM发病到确诊时间	DM确诊后到妇科肿瘤确诊时间	妇科肿瘤确诊方式及症状
1	卵巢癌	2个月	2个月	DM筛查发现
2	卵巢癌	6个月	1年	DM筛查发现,月经不规则1年余
3	卵巢癌	4个月	4个月	DM筛查发现
4	卵巢癌	2年余	2年	DM随访CA12-5升高
5	卵巢癌	1个月	2个月	DM筛查发现
6	宫颈癌	半年	1周	DM筛查发现,发现HPV16阳性2个月余
7	宫颈癌	半个月	1个月	DM筛查发现
8	子宫内膜癌	1个月	1个月	DM筛查发现
9	子宫内膜癌	半年	1个月	DM筛查发现,月经不规则半年余
10	子宫内膜癌	3个月	4个月	DM筛查发现,阴道流血4个月余
11	子宫内膜癌	2月	2个月	绝经后,反复阴道出血3年余

序号	妇科肿瘤	皮疹范围	肌炎表现	肌肉活检	妇科肿瘤治疗方案
1	卵巢癌	额部、眼眶、背部、双上肢、双手指关节伸侧可见红斑,Gottron征阳性	不详	肌炎	手术
2	卵巢癌	全身暗紫红色红斑,颜面部、颈部较重, Gottron征阳性	不详	DM	手术+TC方案化疗
3	卵巢癌	面部,眼睑、躯干下肢可见红斑, Gottron征阳性	无肌痛肌无力	DM	新辅助TC方案化疗+手术
4	卵巢癌	不详	不详	不详	手术+TC方案化疗
5	卵巢癌	额部、颈部、面、耳前、躯干四肢红斑, Gottron征阳性	无肌痛,上肢近端肌力Ⅲ级, 下肢肌力Ⅳ级	请结合临床	手术+TC方案化疗
6	宫颈癌	面部红斑	不详	不详	根治性放化疗
7	宫颈癌	面部、双侧腕关节、颈部、肩部及股沟处红斑, 技工手,Gottron征及披肩征阳性	无肌痛、肌无力	不详	手术
8	子宫内膜癌	颈部、胸前区红斑	无肌痛、肌无力	不详	手术+TC方案化疗+放疗
9	子宫内膜癌	双上眼睑、前胸V字区红斑	四肢肌力Ⅲ级,轻压痛,肌张力正常,环咽肌受累	肌炎	新辅助TC方案化疗+手术
10	子宫内膜癌	面颈部、前胸、后背、腹部、双大腿外侧及双手背红斑,Gottron征阳性	无肌痛、肌无力	DM	手术
11	子宫内膜癌	面部、躯干四肢红斑,部分呈靶形损害, 伴有皮肤破溃	不详	DM	手术+TC方案化疗

序号	妇科肿瘤	抗TIF1-γ抗体		术前AFP (<7 ng/mL)	术前CEA (<5 ng/mL)	术前CA12-5 (<23 U/mL)	术前CA19-9 (<25 U/mL)	术前HE4^a） (pmol/mL)	术前SCCA (<1.5 ng/mL)
序号	妇科肿瘤	MBL法(U/L)	重组蛋白法(U/L)	术前AFP (<7 ng/mL)	术前CEA (<5 ng/mL)	术前CA12-5 (<23 U/mL)	术前CA19-9 (<25 U/mL)	术前HE4^a） (pmol/mL)	术前SCCA (<1.5 ng/mL)
1	卵巢癌	134.1	-	未测	16.47	163.9	61.8	未测	未测
2	卵巢癌	117.9	-	1.92	1.12	823.4	<0.8	未测	未测
3	卵巢癌	77.5	-	5.02	1.52	106.2	4.8	182.1	未测
4	卵巢癌	101.3	-	2.90	1.16	563.8	14.3	414.3	未测
5	卵巢癌	-	71.10	1.76	2.94	224.1	13.0	75.0	未测
6	宫颈癌	-	未测	未测	未测	未测	未测	未测	2.6
7	宫颈癌	-	未测	2.34	1.18	7.6	10.8	未测	5.0
8	子宫内膜癌	-	未测	2.29	2.45	143.1	<0.8	104.0	未测
9	子宫内膜癌	-	222.13	2.35	1.37	9.0	10.2	33.0	未测
10	子宫内膜癌	28.4	-	1 052.30	2.03	24.4	12.1	103.0	未测
11	子宫内膜癌	72.9	-	2.25	1.42	10.7	4.7	70.5	未测