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库特纳肿瘤:关于诊断、治疗及预后的病例系列与叙述性综述

Kuttner's Tumour: A Case Series and Narrative Review on Diagnosis, Management, and Outcomes.

作者信息

Al-Deerawi Zaid, El-Badawi Kamal, Shrivastava Arpan, Barrak Husham

机构信息

Queen Elizabeth Hospital, Birmingham B15 2GW, UK.

Queen Elizabeth the Queen Mother Hospital, Margate, Kent CT9 4AN, UK.

出版信息

J Clin Med. 2025 May 6;14(9):3208. doi: 10.3390/jcm14093208.

DOI:10.3390/jcm14093208
PMID:40364239
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC12072982/
Abstract

: Kuttner's tumour refers to chronic sclerosing sialadenitis commonly affecting the submandibular gland. It clinically mimics submandibular gland neoplasms and has a possible association with IgG4-related disease. The literature on standardised diagnostic and management pathways is limited. This paper presents a case series and narrative literature review to support a proposed diagnostic and management approach for Kuttner's tumour. : Twelve cases of patients with a Kuttner's tumour aged between 55 and 87, identified from 2018 to 2025 through hospital records, were reviewed. All patients underwent ultrasound assessment using standardised diagnostic criteria and were followed up clinically and radiologically every six months. In parallel, we performed a narrative review of studies published between 2005 and 2025, identifying nine relevant articles to contextualise our findings. : Our 12-patient case series highlights the potential for Kuttner's tumour to progress to bilateral involvement and IgG4-related disease. Most cases resolved spontaneously with ultrasound-led monitoring. One progressed to IgG4-RD and responded to glucocorticoids. Our findings support the selective use of invasive tests, baseline serum IgG4 testing, and a six-monthly follow-up strategy. Despite similarities within the existing literature, international variation highlights the need for standardised diagnostic and management protocols. : We recommend a conservative, structured approach to managing Kuttner's tumour, with six-monthly clinical and radiological follow-ups. Based on one case progressing to multi-organ IgG4-related disease, we now recommend routinely measuring serum IgG4 concentrations at diagnosis. The role of magnetic resonance imaging, fine-needle aspiration cytology, and biopsy should be considered on a case-by-case basis. Further research is needed to validate this approach and assess long-term outcomes.

摘要

库特纳瘤是指通常累及下颌下腺的慢性硬化性涎腺炎。其临床表现类似于下颌下腺肿瘤,可能与IgG4相关疾病有关。关于标准化诊断和管理途径的文献有限。本文通过病例系列和叙述性文献综述,以支持提出的库特纳瘤诊断和管理方法。

回顾了2018年至2025年通过医院记录确定的12例年龄在55至87岁之间的库特纳瘤患者。所有患者均按照标准化诊断标准接受超声评估,并每六个月进行临床和影像学随访。同时,我们对2005年至2025年发表的研究进行了叙述性综述,确定了9篇相关文章以阐述我们的研究结果。

我们的12例患者病例系列突出了库特纳瘤进展为双侧受累和IgG4相关疾病的可能性。大多数病例在超声引导监测下自发缓解。1例进展为IgG4相关性疾病并对糖皮质激素有反应。我们的研究结果支持选择性使用侵入性检查、基线血清IgG4检测和每六个月一次的随访策略。尽管现有文献存在相似之处,但国际差异凸显了标准化诊断和管理方案的必要性。

我们建议采用保守、结构化的方法来管理库特纳瘤,每六个月进行临床和影像学随访。基于1例进展为多器官IgG4相关疾病的病例,我们现在建议在诊断时常规测量血清IgG4浓度。磁共振成像、细针穿刺细胞学检查和活检的作用应根据具体情况考虑。需要进一步研究来验证这种方法并评估长期结果。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/66fe/12072982/752dede05cf6/jcm-14-03208-g007.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/66fe/12072982/a2a480448e46/jcm-14-03208-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/66fe/12072982/65fde1e897d3/jcm-14-03208-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/66fe/12072982/56377f432f56/jcm-14-03208-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/66fe/12072982/050da981e5ee/jcm-14-03208-g004.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/66fe/12072982/0449ded9caa4/jcm-14-03208-g005.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/66fe/12072982/f2f902dc76f5/jcm-14-03208-g006.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/66fe/12072982/752dede05cf6/jcm-14-03208-g007.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/66fe/12072982/a2a480448e46/jcm-14-03208-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/66fe/12072982/65fde1e897d3/jcm-14-03208-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/66fe/12072982/56377f432f56/jcm-14-03208-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/66fe/12072982/050da981e5ee/jcm-14-03208-g004.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/66fe/12072982/0449ded9caa4/jcm-14-03208-g005.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/66fe/12072982/f2f902dc76f5/jcm-14-03208-g006.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/66fe/12072982/752dede05cf6/jcm-14-03208-g007.jpg

相似文献

1
Kuttner's Tumour: A Case Series and Narrative Review on Diagnosis, Management, and Outcomes.库特纳肿瘤:关于诊断、治疗及预后的病例系列与叙述性综述
J Clin Med. 2025 May 6;14(9):3208. doi: 10.3390/jcm14093208.
2
Kuttner's tumour (chronic sclerosing sialadenitis) of the submandibular gland: a clinical perspective.下颌下腺库特纳瘤(慢性硬化性涎腺炎):临床视角
Hong Kong Med J. 2008 Feb;14(1):46-9.
3
[Küttner's tumor (chronic sclerosing sialadenitis) - a rare cause of submandibular gland enlargement].[库特纳瘤(慢性硬化性涎腺炎)——下颌下腺肿大的罕见原因]
Otolaryngol Pol. 2011 Jul-Aug;65(4):289-92. doi: 10.1016/S0030-6657(11)70693-X.
4
Küttner's tumour: an unusual cause of salivary gland enlargement.库特纳瘤:唾液腺肿大的罕见病因。
Hippokratia. 2008 Jan;12(1):56-8.
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MRI of chronic sclerosing sialoadenitis.慢性硬化性涎腺炎的磁共振成像
Br J Radiol. 2008 Jul;81(967):531-6. doi: 10.1259/bjr/38960620. Epub 2008 Mar 3.
6
Küttner's tumour of the submandibular gland.库特纳下颌下腺肿瘤。
B-ENT. 2006;2(4):197-9.
7
An unusual presentation of chronic sclerosing sialadenitis of submandibular gland (Kuttner's tumour).下颌下腺慢性硬化性涎腺炎(库特纳瘤)的一种不寻常表现。
BMJ Case Rep. 2019 Aug 28;12(8):e231189. doi: 10.1136/bcr-2019-231189.
8
Mikulicz's disease and Küttner's tumor as manifestations of IgG4-related diseases: a review of the literature.米库利奇病和库特纳瘤作为IgG4相关性疾病的表现:文献综述
Reumatologia. 2020;58(4):243-250. doi: 10.5114/reum.2020.98437. Epub 2020 Aug 31.
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Chronic sclerosing sialadenitis (Küttner's tumor) of the submandibular salivary gland: Our experience of one case report.下颌下涎腺慢性硬化性涎腺炎(库特纳瘤):我们的一例病例报告经验。
Clin Case Rep. 2019 Jul 15;7(8):1600-1604. doi: 10.1002/ccr3.2303. eCollection 2019 Aug.
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Küttner's tumour: a case report.库特纳瘤:一例病例报告。
Acta Chir Belg. 2008 Sep-Oct;108(5):621-4. doi: 10.1080/00015458.2008.11680304.

本文引用的文献

1
Chronic Sclerosing Sialadenitis of the Submandibular Gland and its Histopathological Spectrum in the IgG4-Related Disease: a Series of 17 Cases.慢性下颌下腺硬化性唾液腺炎及其在 IgG4 相关疾病中的组织病理谱:17 例系列病例。
Head Neck Pathol. 2024 May 13;18(1):42. doi: 10.1007/s12105-024-01651-4.
2
Epidemiology, outcomes, and prognostic factors in submandibular gland carcinomas: a national DAHANCA study.颌下腺癌的流行病学、结局和预后因素:一项全国性 DAHANCA 研究。
Eur Arch Otorhinolaryngol. 2023 Jul;280(7):3405-3413. doi: 10.1007/s00405-023-07940-y. Epub 2023 Apr 13.
3
Not All Küttner Tumors Are IgG4-Related Disease (IgG4-RD).
并非所有 Küttner 肿瘤都是 IgG4 相关疾病(IgG4-RD)。
Head Neck Pathol. 2021 Dec;15(4):1322-1327. doi: 10.1007/s12105-020-01268-3. Epub 2021 Jan 4.
4
The 2020 revised comprehensive diagnostic (RCD) criteria for IgG4-RD.2020年修订的IgG4相关性疾病综合诊断(RCD)标准。
Mod Rheumatol. 2021 May;31(3):529-533. doi: 10.1080/14397595.2020.1859710. Epub 2021 Jan 28.
5
Advances in the diagnosis and management of IgG4 related disease.IgG4 相关疾病的诊断与治疗进展。
BMJ. 2020 Jun 16;369:m1067. doi: 10.1136/bmj.m1067.
6
Sclerosing sialadenitis of the submandibular gland is rarely an immunoglobulin G4-related disease in the Finnish population.下颌下腺硬化性唾液腺炎在芬兰人群中很少是 IgG4 相关疾病。
Mod Pathol. 2020 Apr;33(4):551-559. doi: 10.1038/s41379-019-0395-5. Epub 2019 Nov 3.
7
Diagnostic Accuracy of Fine Needle Aspiration Cytology in Lesions of Oral Cavity and Salivary Glands: A Clinico-Pathological Study.细针穿刺细胞学检查在口腔及唾液腺病变中的诊断准确性:一项临床病理研究
Open Dent J. 2018 Sep 28;12:782-790. doi: 10.2174/1745017901814010782. eCollection 2018.
8
Immunoglobulin G4 related chronic sclerosing sialadenitis.免疫球蛋白G4相关性慢性硬化性涎腺炎
J Laryngol Otol. 2015 Mar;129(3):226-31. doi: 10.1017/S0022215115000195.
9
Clinical relevance of Küttner tumour and IgG4-related dacryoadenitis and sialoadenitis.库特纳瘤与IgG4相关性泪腺炎和涎腺炎的临床相关性
Oral Dis. 2015 Mar;21(2):257-62. doi: 10.1111/odi.12259. Epub 2014 Jun 25.
10
Kuttner tumor.库特纳瘤
Bratisl Lek Listy. 2013;114(1):36-8. doi: 10.4149/bll_2013_009.