超声诊断IgG4相关性疾病2例

2019-12-19 邓椀月 陈琴 周青 实用医院临床杂志

病例1,男,35岁。因“反复淋巴结肿大3年余,左眼外凸1年余,腮腺肿大1月余”于2016年3月3日入院。查体:双眼上睑轻度肿大,左侧颈部及双侧腋下扪及多个肿大淋巴结,质中,颈部最大者约1.5 cm×1.5 cm,活动,无压痛;腋下淋巴结最大约1.3 cm×0.9 cm,活动度差。双侧腹股沟可扪及多个肿大淋巴结,最大者约1.8 cm×1.5 cm。右侧腮腺明显肿大,左侧腮腺轻度肿大,无明显压痛。

病例1,男,35岁。因“反复淋巴结肿大3年余,左眼外凸1年余,腮腺肿大1月余”于2016年3月3日入院。查体:双眼上睑轻度肿大,左侧颈部及双侧腋下扪及多个肿大淋巴结,质中,颈部最大者约1.5 cm×1.5 cm,活动,无压痛;腋下淋巴结最大约1.3 cm×0.9 cm,活动度差。双侧腹股沟可扪及多个肿大淋巴结,最大者约1.8 cm×1.5 cm。右侧腮腺明显肿大,左侧腮腺轻度肿大,无明显压痛。
 
CT示:纵膈内及双侧腋窝区见多枚肿大淋巴结。颅脑MRI提示:左侧眼眶外直肌呈肿块改变,左侧上直肌增粗,病变性质待定,双侧眼眶泪腺增大,性质待定。超声检查:双眼泪腺区探及低回声结节,左侧大小约16.8mm×8.4mm,右侧大小约16.3mm×9.7mm,边界尚清,形态不规则,结节内血流信号1级。左眼球后肌锥内靠外侧探及大小约12.9mm×2.8mm低回声结节,边界清楚,形态欠规则,与外直肌分界不清,未见明显血流信号。
 
双侧腮腺实质回声欠均匀;双侧腮腺下极分别探及实性低回声团,左侧大小约18mm×13mm,右侧大小约24mm×15mm,边界清楚,形态不规则,内部回声欠均匀,血流信号1~3级。双侧腮腺内还探及多个淋巴结,左侧大者约11mm×7mm,右侧大者约10mm×6mm,皮质增厚,部分皮髓质分界欠清,内可见1级血流信号(图1)。




图1 IgG4患者浅表超声影像改变a:左侧泪腺区低回声结节,血流信号1级;b:左侧腮腺内肿大淋巴结;c:右侧下腺内肿大淋巴结;d:右侧颈部III区肿大淋巴结
 
注入造影剂后肿块稍早于正常腮腺组织增强,呈整体弥漫性增强,达峰时团块呈均匀高增强,团块周缘可见增强环,增强后肿块边界较清楚,形态不规则,大小未见明显变化,团块内造影剂消退缓慢。超声造影提示右侧腮腺下极实性团块为良性肿瘤性病变。双侧颈部、腋窝、腹股沟探及多个淋巴结回声,颈部最大者为18mm×7mm,腋窝最大者为42mm×18mm,腹股沟最大者为21mm×10mm,皮质增厚,皮髓质分界欠清,内可见1~2级血流信号。胰头稍肿大,未见肿块。
 
实验室检查:白细胞计数10.04×109/L、单核细胞数0.904×109/L、嗜碱性粒细胞数0.132×109/L。IgG418600mg/L(正常值<1350mg/L)。右侧腮腺肿块病理提示:右腮腺浆细胞为CD138(+),IgG4(+)浆细胞大于50个/HPF;生发中心及外套层为CD20(+),散在分布小淋巴细胞CD3ε(+),Ki67阳性率生发中心约为80%;以上结果支持IgG4相关性疾病。
 
病例2,男,67岁。因“发现胰腺占位6天”于2016年6月21日入院。查体:双眼上睑轻度肿大,左下颌区见一长约6 cm手术瘢痕(患者自诉5年前行左下颌包块切除,病检提示良性)。双侧腮腺稍肿大,腹部查体未见异常,全身浅表淋巴结无肿大。CT示两期增强胰腺体尾部强化明显低于正常胰腺组织,强化欠均匀,胰腺周围脂肪浑浊。胰周血管未见明显受损,提示自身免疫性胰腺炎。
 
超声示双侧泪腺稍肿大呈弥漫性不均匀性改变;左眼上直肌增厚,右侧上、下直肌稍增厚;双侧腮腺内探及多个肿大淋巴结,大者约10mm×7mm,右侧颌下腺探及实性低回声团,大者约14mm×13mm,提示良性病变。双侧颈部II区探及多个肿大淋巴结,大者约9mm×8mm,皮髓质分界欠清,皮质稍增厚。胰头肿大呈肿块样,大小约2.9 cm×2.7 cm。注入造影剂后观察胰头与胰体、胰尾同进等增强,提示为非肿瘤性病变,结合泪腺及颌下腺声像改变建议排除IgG4相关性疾病(图2)。实验室检查:血常规未见明显异常,总蛋白60.3g/L、白蛋白36.7g/L,CEA、CA125正常;血清IgG4水平为4340mg/L。患者无自觉症状而放弃治疗。


图2 IgG4患者胰腺超声影像改变a:胰头肿大二维图;b:胰腺造影图,造影后胰头与胰体、尾同进等增强。
 
讨论
 
IgG4相关性疾病是一种与IgG4淋巴细胞密切相关的慢性、系统性疾病。多数患者伴有血浆IgG4水平升高,几乎所有患者均伴有受累器官IgG4阳性的浆细胞浸润,常见受累器官包括泪腺、胰腺和腹膜后间隙等,累及的器官或组织由于慢性炎症及纤维化进程可导致弥漫性肿大。
 
常见累及器官超声表现:
 
①胰腺:自身免疫性胰腺炎常规腹部超声表现为胰腺弥漫性肿大,呈所谓“腊肠样”改变,伴其内弥漫的微小低回声区或局部低回声肿物,类似于胰腺肿瘤的表现。
 
②唾液腺:IgG4相关疾病累及唾液腺常见于Kuttner瘤和Mikulicz病。Kuttner瘤即慢性硬化性唾液腺炎,病变早期,涎腺无明显增大,表现为腺体回声局灶性或弥漫性轻度不均;随着病情进展,由于反复的淋巴细胞浸润,导管周围组织明显纤维化,导管上皮增殖等原因导致腺体增大,回声明显粗糙不均,腺体回声呈“地图状”或“花斑状”,常可见扩张的导管,管壁增厚。病程晚期涎腺由于腺体明显纤维化,腺实质萎缩,腺体质地较硬且缩小。Mikulicz病表现为腮腺或颌下腺弥漫性肿大,质地较硬,表面光滑,与周围组织无粘连,有时也可形成肿瘤样结节。
 
③腹膜后间隙:IgG4相关疾病累及腹膜后组织多导致腹膜后纤维化。该病累及多器官时,影像学如CT、MR也均能很好显示受累病变改变。
 
本报告2例患者均多处淋巴结肿大,应与Castleman病、淋巴瘤、干燥综合征等疾病鉴别。鉴别要点:
 
Castleman病:可发生于全身淋巴结,以腹部及颈部常见。二维超声表现为肿块呈圆形或类圆形,边界清,有包膜,内部回声均匀。此病术前诊断困难,明确诊断需病理检查。
 
淋巴瘤:是淋巴系统常见恶性肿瘤,主要侵犯淋巴结和结外淋巴组织,以颈部淋巴结最常见。超声表现为淋巴结显著增大,呈类圆形或圆形,多数多发。非霍奇金淋巴瘤(NHL,B型细胞为主)内可见点线样回声,为淋巴细胞增生后内部结构移位破坏所致。通过病理可以鉴别。
 
干燥综合征又称舍格综合征,病因不明,常合并结缔组织病、类风湿关节炎等疾病。结合口干、眼干等临床症状可以鉴别,做唇腺活检可诊断。
 
病例2胰头肿大,应与胰腺癌鉴别。目前,IgG4-RD的诊断主要采用2012年1月日本发表的综合诊断标准。主要内容包括:①一或多个器官出现弥漫性/局限性肿胀或肿块的临床表现。②血清IgG4浓度≥1350mg/L。③组织病理学检查:显著的淋巴细胞、浆细胞浸润和纤维化;IgG4阳性浆细胞浸润:IgG4阳性/IgG阳性细胞>40%,且IgG4阳性浆细胞>10个高倍视野。①、②、③全满足便确诊为此病,①、③满足很可能为此病,只满足①、②可能为此病。例1三条全满足,因此确诊为IgG4相关性疾病;例2满足前两条,因此可能为此病。
 
超声在诊断及鉴别诊断IgG4相关性疾病中可发挥重要作用。然而该病无特征性的超声表现。超声若发现多部位病变,应警惕此病,结合临床表现、其他影像学检查、血清学检查可初步诊断,明确诊断需病理检查。
 
原始出处:

邓椀月,陈琴,周青,吴昊,邱金鸾,李敏.超声诊断IgG4相关性疾病2例报告[J].实用医院临床杂志,2018(01):214-215.

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  3. [GetPortalCommentsPageByObjectIdResponse(id=1837769, encodeId=0a63183e76920, content=<a href='/topic/show?id=3f34e253206' target=_blank style='color:#2F92EE;'>#相关性#</a>, beContent=null, objectType=article, channel=null, level=null, likeNumber=27, replyNumber=0, topicName=null, topicId=null, topicList=[TopicDto(id=72532, encryptionId=3f34e253206, topicName=相关性)], attachment=null, authenticateStatus=null, createdAvatar=, createdBy=a9de34, createdName=shenxlcyp, createdTime=Fri Jun 26 23:24:00 CST 2020, time=2020-06-26, status=1, ipAttribution=), GetPortalCommentsPageByObjectIdResponse(id=1350154, encodeId=9b1c1350154f5, content=<a href='/topic/show?id=0ee1951250' target=_blank style='color:#2F92EE;'>#IgG4相关性疾病#</a>, beContent=null, objectType=article, channel=null, level=null, likeNumber=40, replyNumber=0, topicName=null, topicId=null, topicList=[TopicDto(id=9512, encryptionId=0ee1951250, topicName=IgG4相关性疾病)], attachment=null, authenticateStatus=null, createdAvatar=, createdBy=4c37265, createdName=zhaojie88, createdTime=Sat Dec 21 12:24:00 CST 2019, time=2019-12-21, status=1, ipAttribution=), GetPortalCommentsPageByObjectIdResponse(id=1482976, encodeId=0a3014829e69c, content=<a href='/topic/show?id=5d52950839' target=_blank style='color:#2F92EE;'>#IgG4#</a>, beContent=null, objectType=article, channel=null, level=null, likeNumber=38, replyNumber=0, topicName=null, topicId=null, topicList=[TopicDto(id=9508, encryptionId=5d52950839, topicName=IgG4)], attachment=null, authenticateStatus=null, createdAvatar=, createdBy=bcbb7863836, createdName=ms5106640509429053, createdTime=Sat Dec 21 12:24:00 CST 2019, time=2019-12-21, status=1, ipAttribution=), GetPortalCommentsPageByObjectIdResponse(id=1560009, encodeId=62471560009c5, content=<a href='/topic/show?id=c1eb9282699' target=_blank style='color:#2F92EE;'>#超声诊断#</a>, beContent=null, objectType=article, channel=null, level=null, likeNumber=37, replyNumber=0, topicName=null, topicId=null, topicList=[TopicDto(id=92826, encryptionId=c1eb9282699, topicName=超声诊断)], attachment=null, authenticateStatus=null, createdAvatar=null, createdBy=6ebf15056071, createdName=tcm104hq, createdTime=Sat Dec 21 12:24:00 CST 2019, time=2019-12-21, status=1, ipAttribution=), GetPortalCommentsPageByObjectIdResponse(id=1566018, encodeId=e803156601831, content=<a href='/topic/show?id=c1eb9282699' target=_blank style='color:#2F92EE;'>#超声诊断#</a>, beContent=null, objectType=article, channel=null, level=null, likeNumber=40, replyNumber=0, topicName=null, topicId=null, topicList=[TopicDto(id=92826, encryptionId=c1eb9282699, topicName=超声诊断)], attachment=null, authenticateStatus=null, createdAvatar=null, createdBy=6ebf15056071, createdName=tcm104hq, createdTime=Sat Dec 21 12:24:00 CST 2019, time=2019-12-21, status=1, ipAttribution=), GetPortalCommentsPageByObjectIdResponse(id=1615207, encodeId=5141161520ef8, content=<a href='/topic/show?id=010c9506a3' target=_blank style='color:#2F92EE;'>#IgG#</a>, beContent=null, objectType=article, channel=null, level=null, likeNumber=34, replyNumber=0, topicName=null, topicId=null, topicList=[TopicDto(id=9506, encryptionId=010c9506a3, topicName=IgG)], attachment=null, authenticateStatus=null, createdAvatar=null, createdBy=6c4f19588882, createdName=ms5768352621950538, createdTime=Sat Dec 21 12:24:00 CST 2019, time=2019-12-21, status=1, ipAttribution=), GetPortalCommentsPageByObjectIdResponse(id=377205, encodeId=09e33e720518, content=超声诊断这个有难度。, beContent=null, objectType=article, channel=null, level=null, likeNumber=83, replyNumber=0, topicName=null, topicId=null, topicList=[], attachment=null, authenticateStatus=null, createdAvatar=, createdBy=43a21971117, createdName=njwbhuang, createdTime=Fri Dec 20 10:56:38 CST 2019, time=2019-12-20, status=1, ipAttribution=)]
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    2019-12-20 njwbhuang

    超声诊断这个有难度。

    0

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