原发口腔外周T细胞淋巴瘤非特指型的特点分析

doi:10.3969/j.issn.1674-8115.2025.05.015

上海交通大学学报（医学版） ›› 2025, Vol. 45 ›› Issue (5): 653-660.doi: 10.3969/j.issn.1674-8115.2025.05.015

• 短篇论著 • 上一篇

原发口腔外周T细胞淋巴瘤非特指型的特点分析

黄润语¹, 张春叶², 张颖¹, 赵峥岩¹, 杨扬³, 吴岚¹()

^1.上海交通大学医学院附属第九人民医院口腔黏膜病科，上海交通大学口腔医学院，国家口腔医学中心，国家口腔疾病临床医学研究中心，上海市口腔医学重点实验室，上海市口腔医学研究所，上海 200011
^2.上海交通大学医学院附属第九人民医院口腔病理科，上海交通大学口腔医学院，国家口腔医学中心，国家口腔疾病临床医学研究中心，上海市口腔医学重点实验室，上海市口腔医学研究所，上海 200011
^3.徐州医科大学附属口腔医院牙周黏膜病科，徐州 221002

收稿日期:2024-11-07 接受日期:2025-02-24 出版日期:2025-05-28 发布日期:2025-05-28
通讯作者: 吴　岚，主任医师，博士；电子信箱：teana_wu@sina.com。
作者简介:黄润语（1999—），女，住院医师，硕士生；电子信箱：shdiver@outlook.com。
基金资助:
上海市科学技术委员会基金(21S21902000);上海市卫生健康委员会项目［（ZY（2021-2023）-0207-01-04,2022CX009）］;上海交通大学医学院附属第九人民医院交叉基金(JYJC202111)

Features of oral peripheral T-cell lymphoma, not otherwise specified

HUANG Runyu¹, ZHANG Chunye², ZHANG Ying¹, ZHAO Zhengyan¹, YANG Yang³, WU Lan¹()

^1.Department of Oral Mucosal Diseases, Shanghai Ninth People's Hospital, Shanghai Jiao Tong University School of Medicine; College of Stomatology, Shanghai Jiao Tong University; National Center for Stomatology; National Clinical Research Center for Oral Diseases; Shanghai Key Laboratory of Stomatology; Shanghai Research Institute of Stomatology, Shanghai 200011, China
^2.Department of Oral Pathology, Shanghai Ninth People's Hospital, Shanghai Jiao Tong University School of Medicine; College of Stomatology, Shanghai Jiao Tong University; National Center for Stomatology; National Clinical Research Center for Oral Diseases; Shanghai Key Laboratory of Stomatology; Shanghai Research Institute of Stomatology, Shanghai 200011, China
^3.Department of Periodontal and Mucosal Diseases, Affiliated Stomatological Hospital of Xuzhou Medical University, Xuzhou 221002, China.

Received:2024-11-07 Accepted:2025-02-24 Online:2025-05-28 Published:2025-05-28
Contact: WU Lan, E-mail: teana_wu@sina.com.
Supported by:
Shanghai Municipal Commission of Science and Technology Fund(21S21902000);Shanghai Municipal Commission of Health Fund [(ZY(2021-2023)- 0207-01-04, 2022CX009)];Cross Project Cooperation Fund of Shanghai Ninth People′s Hospital, Shanghai Jiao Tong University School of Medicine(JYJC202111)

摘要/Abstract

摘要：

目的·探讨原发于口腔的外周T细胞淋巴瘤-非特指型（peripheral T-cell lymphoma， not otherwise specified，PTCL-NOS）的临床表现和免疫表型特征。方法·回顾性分析2020年8月至2024年8月就诊于上海交通大学医学院附属第九人民医院口腔黏膜病科、口腔为首发部位、确诊为PTCL-NOS的患者的病史和病理资料，并检索PubMed、Web of Science、Embase、Scopus和中国知网共5个数据库，结合2014年1月至2024年9月国际上相关病例报道，对临床特点、病理学特点进行分析归纳。结果·共20例口腔PTCL-NOS病例纳入研究。其中男性11例（55.0%），女性9例（45.0%）；患者首诊年龄25~77岁，平均（52.53±12.94）岁；好发部位为舌部（25.0%）、腭部（25.0%）及颊部（20.0%）；19例（95.0%）不伴B症状。纳入病例免疫表型阳性表达情况为CD3（19/19）、CD4（11/13）、CD8（7/12）、CD2（5/6）、CD7（5/5）、TIA-1（6/7）、GB（9/13）、perforin（4/6），EBER均阴性表达（8/8）。85%的病例Ki-67增殖指数≥60%。结论·口腔PTCL-NOS极为罕见，临床进展极具侵袭性，口腔表现为深而大的黏膜溃疡，基底高低不平，可扪及结节。病理特征具有异质性，免疫表型检测有助于早期诊断和分型。口腔科医师需加深对该疾病的认识，以避免错过此类恶性疾病的早期诊断与治疗。

关键词: 口腔特征, 外周T细胞淋巴瘤, 非特指型, 临床病理

Abstract:

Objective ·To investigate the clinical manifestations and immunophenotypic features of peripheral T-cell lymphoma, not otherwise specified (PTCL-NOS), involving the oral cavity. Methods ·The medical histories and pathology records of patients diagnosed with oral PTCL-NOS in the Department of Oral Mucosal Diseases of Shanghai Ninth People′s Hospital, Shanghai Jiao Tong University School of Medicine, between August 2020 and August 2024 were retrospectively analyzed. In addition, 5 databases, including PubMed, Web of Science, Embase, Scopus, and CNKI, were searched, and relevant cases reported internationally from January 2014 to September 2024 were reviewed. Results ·A total of 20 oral PTCL-NOS cases were included, comprising 11 males (55.0%) and 9 females (45.0%). The patients′ ages at initial diagnosis ranged from 25 to 77 years, with a mean age of (52.53±12.94) years. The most common sites were the tongue (25.0%), palate (25.0%), and buccal mucosa (20.0%). Nineteen cases (95.0%) had no B symptoms. The cases were positive for CD3 (19/19), CD4 (11/13), CD8 (7/12), CD2 (5/6), CD7 (5/5), TIA-1 (6/7), GB (9/13), perforin (4/6). EBER expression was negative (8/8). The Ki-67 proliferation index was ≥60% in 85% of cases. Conclusions ·Oral PTCL-NOS is extremely rare and has an aggressive clinical behavior. The oral manifestation presents as deep and large mucosal ulcers with uneven bases, and nodules can be palpable. The pathological features are heterogeneous. Immunophenotype detection is useful for early diagnosis and classification. It is essential for stomatologists to enhance their awareness of this malignancy to avoid delayed diagnosis and treatment.

Key words: oral feature, peripheral T-cell lymphoma (PTCL), not otherwise specified (NOS), clinical pathology

中图分类号:

R733.4

黄润语, 张春叶, 张颖, 赵峥岩, 杨扬, 吴岚. 原发口腔外周T细胞淋巴瘤非特指型的特点分析[J]. 上海交通大学学报（医学版）, 2025, 45(5): 653-660.

HUANG Runyu, ZHANG Chunye, ZHANG Ying, ZHAO Zhengyan, YANG Yang, WU Lan. Features of oral peripheral T-cell lymphoma, not otherwise specified[J]. Journal of Shanghai Jiao Tong University (Medical Science), 2025, 45(5): 653-660.

图/表 6

参考文献 22

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Year	Patient/ reference	Gender/ age	Location	B symptom	Radiographic feature	Ann Arbor staging	Treatment
2023	Case 1	F/55	Tongue	None	MRI showed a vaguely-bordered soft tissue thickening at the anterior tongue. In addition, MRI showed an intermediate T1 signal and slightly high T2 signal lesion at the right side of the tongue, which was about 3.6 cm×1.3 cm. The lesion demonstrated heterogeneous contrast enhancement	Ⅰ EA	/
2024	Case 2	M/44	Hard palate	None	MRI illustrated a vaguely-bordered soft tissue thickening, which measured approximately 3.4 cm×2.7 cm×1.2 cm and showed a slightly low T1 signal and slightly high T2 signal. The lesion demonstrated heterogeneous contrast enhancement. There were several enlarged lymph nodes in the level Ⅱ‒level Ⅳ cervical	Ⅳ A	GemOx-D
2014	1/[2]	M/59	The right side of the tongue base	None	CT scan presented an ill-defined, heterogeneous enhancing soft tissue mass lesion in the right tongue base and enlarged lymph nodes in the right level Ⅱ	Ⅱ A	CHOP+RT+VMAT
2016	2/[3]	F/50	The left side of the anterior tongue	None	MRI T2WI showed a bordered lesion at the anterior tongue	Ⅰ	None
2017	3/[4]	F/42	Near the median sulcus of the tongue	None	Enhanced MRI showed a bordered, bar-shaped, irregular lesion with intermediate T1 signal and slightly high T2 signal in the right portion of the tongue, and its size was about 8.5 mm×24.5 mm	/	Surgery, CHOP
2018	4/[5]	F/25	Upper lip, palate, and maxillary sinus	Fever, weight loss	CT scan with contrast showed an expansive and infiltrative formation with irregular contours and ulceration, extending from the upper lip to the nasolabial sulcus, with infiltration into the epidermis and nasal mucosa. The formation affected the right tear duct without bone involvement. In addition, CT showed a bilateral increase in the number and size of the cervical lymph nodes, mainly the submandibular chain	Ⅳ B	CHOEP
2020	5/[6]	M/ 75‒80	The right side of the tongue base	None	CT scan showed a well-bordered cystic mass (2 cm in diameter) at the right base of the tongue extending into the pharynx	Ⅳ A	GDP+CHOP
2021	6/[7]	F/63	Submandibular region	/	/	/	/
2021	7/[7]	M/67	Buccal mucosa	/	/	/	/
2021	8/[7]	M/57	Hard palate	/	/	/	/
2021	9/[7]	M/38	Soft palate	/	/	/	/
2021	10/[7]	M/59	Buccal mucosa	/	/	/	/
2021	11/[7]	F/56	Tonsil	/	/	/	/
2021	12/[7]	F/66	Tonsil	/	/	/	/
2023	13/[8]	M/34	Right buccal mucosa	None	CT scan imaging showed destructive and expansive mass with central necrosis, extending to maxillary and ethmoid sinuses	Ⅱ EA	CHOEP+RT
2024	14/[9]	F/37	Maxillary gingiva	None	/	/	Anthracycline-based regimens
2024	15/[9]	M/44	Buccal mucosa	None	/	/	/
2024	16/[9]	F/62	Palate and gingiva	None	/	/	Anthracycline-based regimens
2024	17/[9]	M/55	Palate and maxillary sinus	None	CT scan illustrated the neoplasm invading the maxillary sinus and the orbital cavity, and extending toward the base of the skull	/	Anthracycline-based regimens
2024	18/[9]	M/61	Soft palate and tonsil	None	/	/	/

Year	Patient/ reference	Gender/ age	Location	B symptom	Radiographic feature	Ann Arbor staging	Treatment
2023	Case 1	F/55	Tongue	None	MRI showed a vaguely-bordered soft tissue thickening at the anterior tongue. In addition, MRI showed an intermediate T1 signal and slightly high T2 signal lesion at the right side of the tongue, which was about 3.6 cm×1.3 cm. The lesion demonstrated heterogeneous contrast enhancement	Ⅰ EA	/
2024	Case 2	M/44	Hard palate	None	MRI illustrated a vaguely-bordered soft tissue thickening, which measured approximately 3.4 cm×2.7 cm×1.2 cm and showed a slightly low T1 signal and slightly high T2 signal. The lesion demonstrated heterogeneous contrast enhancement. There were several enlarged lymph nodes in the level Ⅱ‒level Ⅳ cervical	Ⅳ A	GemOx-D
2014	1/[2]	M/59	The right side of the tongue base	None	CT scan presented an ill-defined, heterogeneous enhancing soft tissue mass lesion in the right tongue base and enlarged lymph nodes in the right level Ⅱ	Ⅱ A	CHOP+RT+VMAT
2016	2/[3]	F/50	The left side of the anterior tongue	None	MRI T2WI showed a bordered lesion at the anterior tongue	Ⅰ	None
2017	3/[4]	F/42	Near the median sulcus of the tongue	None	Enhanced MRI showed a bordered, bar-shaped, irregular lesion with intermediate T1 signal and slightly high T2 signal in the right portion of the tongue, and its size was about 8.5 mm×24.5 mm	/	Surgery, CHOP
2018	4/[5]	F/25	Upper lip, palate, and maxillary sinus	Fever, weight loss	CT scan with contrast showed an expansive and infiltrative formation with irregular contours and ulceration, extending from the upper lip to the nasolabial sulcus, with infiltration into the epidermis and nasal mucosa. The formation affected the right tear duct without bone involvement. In addition, CT showed a bilateral increase in the number and size of the cervical lymph nodes, mainly the submandibular chain	Ⅳ B	CHOEP
2020	5/[6]	M/ 75‒80	The right side of the tongue base	None	CT scan showed a well-bordered cystic mass (2 cm in diameter) at the right base of the tongue extending into the pharynx	Ⅳ A	GDP+CHOP
2021	6/[7]	F/63	Submandibular region	/	/	/	/
2021	7/[7]	M/67	Buccal mucosa	/	/	/	/
2021	8/[7]	M/57	Hard palate	/	/	/	/
2021	9/[7]	M/38	Soft palate	/	/	/	/
2021	10/[7]	M/59	Buccal mucosa	/	/	/	/
2021	11/[7]	F/56	Tonsil	/	/	/	/
2021	12/[7]	F/66	Tonsil	/	/	/	/
2023	13/[8]	M/34	Right buccal mucosa	None	CT scan imaging showed destructive and expansive mass with central necrosis, extending to maxillary and ethmoid sinuses	Ⅱ EA	CHOEP+RT
2024	14/[9]	F/37	Maxillary gingiva	None	/	/	Anthracycline-based regimens
2024	15/[9]	M/44	Buccal mucosa	None	/	/	/
2024	16/[9]	F/62	Palate and gingiva	None	/	/	Anthracycline-based regimens
2024	17/[9]	M/55	Palate and maxillary sinus	None	CT scan illustrated the neoplasm invading the maxillary sinus and the orbital cavity, and extending toward the base of the skull	/	Anthracycline-based regimens
2024	18/[9]	M/61	Soft palate and tonsil	None	/	/	/

Item	Oral squamous cell carcinoma	Major recurrent aphthous ulcer	Traumatic ulceration	Tuberculous ulcer	PTCL-NOS	ENKTL-NT
Population	Middle-aged and elderly people	Adult	Teenagers and elderly people	/	Middle-aged people	Middle-aged people
Oral clinical manifestations	Persistent ulceration with raised margins and necrotic base; indurated on palpation	Oval or round ulcers with a white or yellow pseudomembrane and a surrounding erythematous halo; 1 cm or more in diameter	Frank ulceration is surrounded by a white hyperkeratotic lesion. The location and the shape of the ulcer correspond to the stimulating factor	Deep ulcer with undermined edges and red granular base covered by septic secretion	Ulceration, mass, or necrosis	Necrotic ulceration
Location of lesions	The margin of the tongue, and the belly of the tongue	The non-keratinized and keratinized oral mucosa	In a trauma-prone site	Labial mucosa, vestibule, and tongue	Tongue, palate, and buccal mucosa	Median mucosa of the palate
Systemic signs	Lymph node swelling, pain, or weight loss	/	/	Cough, anorexia, fatigue, low-grade fever and weight loss	Lymph node swelling, extra-nodal involvement of the gastrointestinal tract and skin, anemia, eosinophilia, or hypergammaglobulinaemia	Nasal mucosal symptoms or B symptoms
Clinical course	Persistent	Self-limiting, recurrent, and periodic	Self-limiting	Infection	Aggressive	Aggressive
Complementary examinations	Biopsy	/	/	Chest X-ray, tuberculin skin test, bacterial culturing	Biopsy, blood test, bone marrow biopsy	Biopsy, blood test, bone marrow biopsy, EBER-ISH

Item	Oral squamous cell carcinoma	Major recurrent aphthous ulcer	Traumatic ulceration	Tuberculous ulcer	PTCL-NOS	ENKTL-NT
Population	Middle-aged and elderly people	Adult	Teenagers and elderly people	/	Middle-aged people	Middle-aged people
Oral clinical manifestations	Persistent ulceration with raised margins and necrotic base; indurated on palpation	Oval or round ulcers with a white or yellow pseudomembrane and a surrounding erythematous halo; 1 cm or more in diameter	Frank ulceration is surrounded by a white hyperkeratotic lesion. The location and the shape of the ulcer correspond to the stimulating factor	Deep ulcer with undermined edges and red granular base covered by septic secretion	Ulceration, mass, or necrosis	Necrotic ulceration
Location of lesions	The margin of the tongue, and the belly of the tongue	The non-keratinized and keratinized oral mucosa	In a trauma-prone site	Labial mucosa, vestibule, and tongue	Tongue, palate, and buccal mucosa	Median mucosa of the palate
Systemic signs	Lymph node swelling, pain, or weight loss	/	/	Cough, anorexia, fatigue, low-grade fever and weight loss	Lymph node swelling, extra-nodal involvement of the gastrointestinal tract and skin, anemia, eosinophilia, or hypergammaglobulinaemia	Nasal mucosal symptoms or B symptoms
Clinical course	Persistent	Self-limiting, recurrent, and periodic	Self-limiting	Infection	Aggressive	Aggressive
Complementary examinations	Biopsy	/	/	Chest X-ray, tuberculin skin test, bacterial culturing	Biopsy, blood test, bone marrow biopsy	Biopsy, blood test, bone marrow biopsy, EBER-ISH

Year	Patient/ reference	EBER	CD3	CD4	CD8	CD2	CD7	CD20	Ki-67	Others
2023	Case 1	‒	+	+	+	+	+	‒	60%‒70%	TIA-1 (partial+), GB (partial+), perforin (few cells+)
2024	Case 2	/	+	/	/	+	+	‒	>90%	TIA-1(+), GB(+), perforin(+)
2014	1/[2]	‒	+	+	+	/	/	‒	80%	TIA-1(+), GB(+)
2016	2/[3]	/	+	+	+	Few cells+	Few cells+	‒	30%‒40%	/
2017	3/[4]	‒	+	‒	+	/	/	‒	30%	TIA-1(+)
2018	4/[5]	‒	+	+	‒	+	/	‒	60%	/
2020	5/[6]	‒	+	+	‒	/	/	‒	60%	TIA-1(‒), GB(‒)
2020	6/[7]	‒	+	+	‒	/	/	‒	60%‒95%	LCA(+)
2020	7/[7]	‒	+	/	/	/	/	‒	20%	LCA(+), GB(‒), CD30(‒)
2020	8/[7]	‒	+	+	/	/	/	‒	60%‒95%
2020	9/[7]	/	+	/	/	/	/	‒	60%‒95%	GB(+)
2020	10/[7]	/	/	/	/	/	/	/	60%‒95%	CD30(+)
2020	11/[7]	/	+	+	+	‒	/	‒	60%‒95%	TIA-1(+), GB(+), perforin(+)
2020	12/[7]	/	+	+	+	+	+	‒	60%‒95%	TIA-1(+), GB(+), perforin(+), CD30(‒), PD-1(+)
2023	13/[8]	/	+	/	/	/	+	‒	70%‒80%	CD30(‒)
2024	14/[9]	/	+	/	/	/	/	/	70%	Perforin(‒), GB(‒)
2024	15/[9]	/	+	/	/	/	/	/	80%	GB(‒)
2024	16/[9]	/	+	‒	+	/	/	/	95%	Perforin(‒), GB(+), CD30(‒)
2024	17/[9]	/	+	+	‒	/	/	/	95%	GB(+), CD30(‒)
2024	18/[9]	/	+	+	‒	/	/	/	90%	GB(+), CD30(+)

原发口腔外周T细胞淋巴瘤非特指型的特点分析

Features of oral peripheral T-cell lymphoma, not otherwise specified

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[1]	何嘉音, 陈思远, 施晴, 张慕晨, 易红梅, 董磊, 钱樱, 王黎, 程澍, 许彭鹏, 赵维莅. 肾上腺累及的弥漫性大B细胞淋巴瘤患者临床病理特征、基因突变谱及预后分析[J]. 上海交通大学学报（医学版）, 2025, 45(9): 1194-1201.
[2]	杜沚珊, 王玥, 石子旸, 施晴, 易红梅, 董磊, 王黎, 程澍, 许彭鹏, 赵维莅. 甲状腺弥漫性大B细胞淋巴瘤临床病理特征、基因突变谱及预后分析[J]. 上海交通大学学报（医学版）, 2024, 44(1): 64-71.
[3]	朱田雨, 宗春燕, 许诗琼, 葛盛芳, 范先群, 贾仁兵. 结膜黑色素瘤组织病理学特征、Ki-67表达和预后的关系[J]. 上海交通大学学报（医学版）, 2022, 42(5): 617-623.
[4]	邢正文, 吴滢, 王雪莉, 王庆煜, 王文婷, 李治, 张彬, 金晶. 8例儿童卵巢颗粒细胞瘤临床病理特征及预后分析[J]. 上海交通大学学报(医学版), 2022, 42(2): 192-196.
[5]	叶欣，周晓云，杨莉，王杰，何奇. 不同年龄范围界定下的年轻乳腺癌患者的临床病理特征及预后分析[J]. 上海交通大学学报（医学版）, 2020, 40(3): 351-.
[6]	赵玲，高润霖，刘强，沈艳莹. 5例甲状腺内胸腺癌临床病理分析[J]. 上海交通大学学报（医学版）, 2018, 38(10): 1272-.
[7]	侯悦，李少波，师晓琴，傅国辉，伍均. 结直肠癌KRAS 、NRAS 和BRAF 基因突变与临床病理特征的回顾性分析[J]. 上海交通大学学报（医学版）, 2018, 38(1): 4-.
[8]	施永恒,龙满美,刘强,陈晓宇 . 9 例成人横纹肌肉瘤的临床病理学分析[J]. 上海交通大学学报（医学版）, 2017, 37(7): 1015-.
[9]	刘华，建方方，宋玮，沈立翡 . 绝经前后子宫内膜癌的临床及病理特征分析[J]. 上海交通大学学报（医学版）, 2017, 37(12): 1670-.
[10]	周淳，周曾同，张珺晔，郑元俐. 口腔鳞状细胞乳头状瘤和乳头状癌的临床病理学分析[J]. 上海交通大学学报（医学版）, 2016, 36(4): 550-.
[11]	赫超, 沈艳莹, 倪醒之, 等. 12例胃神经鞘瘤的临床病理特征及预后[J]. , 2012, 32(10): 1343-.