肝脏血管周上皮样细胞肿瘤合并贫血一例并文献复习

doi:10.3877/cma.j.issn.2095-3232.2024.05.021

目的

探讨肝脏血管周上皮样细胞肿瘤（PEComa）诊治及疗效。

方法

回顾哈尔滨医科大学附属第一医院收治1例肝PEComa患者的临床表现、影像学表现、组织病理表现及诊治经过，并文献综述。患者女，46岁，主诉"间断性腹胀1个月"于2022年1月19日入院。查体腹部无阳性体征。肝功能无异常，血常规检查：红细胞计数2.43×10¹²/L，示贫血。肝脏CT检查考虑肝内多发富血供占位，部分囊变，不除外恶性肿瘤。肾脏和肺CT平扫检查发现双肾有多发混杂密度，双肺有多发囊性病变。患者已签署知情同意书，符合医学伦理学规定。

结果

血液内科、风湿免疫科会诊，初步诊断贫血与肝PEComa有关。为进一步明确诊断，行PET-CT检查示肝脏部分病灶内有血肿存在。于2022年1月25日患者行肝左外叶切除、肝右叶部分切除和胆囊切除手术。手术顺利完成，患者术后恢复良好，无发热、腹痛，于术后5 d出院。病理诊断为肝PEComa,细胞异型性不明显，核分裂少见，局部可见坏死；免疫组化检测HMB-45（+），MelanA（弱+），SMA（灶+），Ki-67 5%（+），CD34（血管+），CD68（弱+）。截止至投稿日期，术后18个月无复发。

结论

肝PEComa是一罕见病，临床无典型特征，诊断主要依靠病理学诊断。手术治疗作为治疗首选，术后应重视随访。

关键词: 肝肿瘤, 肝脏, 血管周上皮样细胞肿瘤, 贫血, 外科手术

Objective

To investigate the diagnosis, treatment and clinical efficacy of perivascular epithelioid cell tumor (PEComa) of the liver.

Methods

Clinical characteristics, imaging manifestations, histopathological findings, diagnosis and treatment of a case of PEComa of the liver admitted to the First Affiliated Hospital of Harbin Medical University were retrospectively analyzed, and literature review was performed. The 46-year-old female patient reported chief complaint of "intermittent abdominal distension for 1 month" and was admitted to our hospital on January 19, 2022. No positive signs were found in abdominal examination. No abnormality was found in liver function. Routine blood test: red blood cell count was 2.43×10¹²/L, indicating the sign of anemia. CT scan of the liver prompted multiple space-occupying lesions with abundant blood supply in the liver, accompanied with partial cystic changes. The possibility of malignant tumors could not be excluded. Plain CT scan of the kidney and lung showed multiple mixed densities in bilateral kidneys and multiple cystic lesions in both lungs. The informed consents of the patient was obtained and the local ethical committee approval was received.

Results

After consultation by Department of Hematology and Department of Rheumatology Immunology, preliminary diagnosis suggested that anemia was correlated with PEComa of the liver. To further confirm the diagnosis, PET-CT examination showed hematoma in partial liver lesions. On January 25, 2022, the patient underwent left lateral lobe resection, partial right lobe resection and cholecystectomy. The operation was successfully completed, and the patient was properly recovered without fever or abdominal pain. She was discharged at postoperative 5 d. Pathological diagnosis indicated PEComa of the liver with slight cell atypia, rare mitosis and local necrosis. Immunohistochemical staining revealed HMB-45(+), MelanA (weakly +), SMA (lesions +), Ki-67 5%(+), CD34 (vessels +) and CD68 (weakly +). As of the submission date, no recurrence has been reported at postoperative 18 months.

Conclusions

PEComa of the liver is a rare condition with non-typical clinical features. The diagnosis mainly depends on pathological examination. Surgery is the optimal solution. Extensive attention should be paid to postoperative follow-up.

Key words: Liver neoplasms, Liver, Perivascular epithelioid cell carcinoma (PEComa), Anemia, Surgical procedures, operative

图1 一例肝脏血管周上皮样细胞肿瘤患者CT检查注：a为平扫肝左外叶和右叶有3个较大的团块，边界清晰，密度不均，左叶病灶较大，内部可看到囊状低密度影；b为动脉期实质部分强化；c为门静脉期轻度强化；d为延迟期略低于肝实质强化；e为肺部CT平扫示双肺有多发囊性病变；f为肾脏CT平扫示双肾多发混杂密度

图2 一例肝脏血管周上皮样细胞肿瘤患者病理学检查注：HE染色见成熟的脂肪、血管和上皮样梭形细胞（×200）

表1 表现为自发性破裂的肝PEComa病例

作者	年份	性别	年龄	肿瘤大小（cm）	症状	治疗
Huber等^[6]	1996	女	22	8	失血性休克	急诊切除术
Guidi等^[7]	1997	男	74	10	上腹部疼痛	急诊切除术
Tsui等^[8]	1999	男	56	6.5	未记录	急诊切除术
Zhou等^[9]	2008	未记录	未记录	5	失血性休克	急诊剖腹止血
Ding等^[10]	2011	女	56	6	未记录	急诊剖腹止血后择期切除术
Occhionorelli等^[11]	2013	女	25	9	上腹部疼痛	急诊剖腹止血后择期切除术
Tajima等^[12]	2014	男	38	10.5	上腹部疼痛	急诊剖腹止血后择期切除术
Aoki等^[13]	2014	女	70	7	右背痛	动脉栓塞后择期切除术
Kai等^[14]	2015	女	77	2.5	失血性休克	动脉栓塞后选择性切除
陈柏柱等^[15]	2015	男	49	3	上腹部疼痛	急诊剖腹止血
Kim等^[16]	2017	男	31	12.5	上腹部疼痛	动脉栓塞后选择性切除
D'Silva等^[17]	2021	女	47	19	右侧腹部疼痛肿胀	动脉栓塞后选择性切除
陈凯等^[18]	2021	男	69	17×14×9	右上腹持续性隐痛伴腹胀1周	限期选择性切除术
Velasco-Albendea等^[19]	2023	女	35	8	缺乏食欲，体重减轻，乏力，全身不适，消化道不适	随访2个月后行选择性切除术
沈强等^[20]	2023	男	31	8.5×6.5	上腹部疼痛	未记录

表1 表现为自发性破裂的肝PEComa病例

作者	年份	性别	年龄	肿瘤大小（cm）	症状	治疗
Huber等^[6]	1996	女	22	8	失血性休克	急诊切除术
Guidi等^[7]	1997	男	74	10	上腹部疼痛	急诊切除术
Tsui等^[8]	1999	男	56	6.5	未记录	急诊切除术
Zhou等^[9]	2008	未记录	未记录	5	失血性休克	急诊剖腹止血
Ding等^[10]	2011	女	56	6	未记录	急诊剖腹止血后择期切除术
Occhionorelli等^[11]	2013	女	25	9	上腹部疼痛	急诊剖腹止血后择期切除术
Tajima等^[12]	2014	男	38	10.5	上腹部疼痛	急诊剖腹止血后择期切除术
Aoki等^[13]	2014	女	70	7	右背痛	动脉栓塞后择期切除术
Kai等^[14]	2015	女	77	2.5	失血性休克	动脉栓塞后选择性切除
陈柏柱等^[15]	2015	男	49	3	上腹部疼痛	急诊剖腹止血
Kim等^[16]	2017	男	31	12.5	上腹部疼痛	动脉栓塞后选择性切除
D'Silva等^[17]	2021	女	47	19	右侧腹部疼痛肿胀	动脉栓塞后选择性切除
陈凯等^[18]	2021	男	69	17×14×9	右上腹持续性隐痛伴腹胀1周	限期选择性切除术
Velasco-Albendea等^[19]	2023	女	35	8	缺乏食欲，体重减轻，乏力，全身不适，消化道不适	随访2个月后行选择性切除术
沈强等^[20]	2023	男	31	8.5×6.5	上腹部疼痛	未记录

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Perivascular epithelioid cell tumor of the liver complicated with anemia: a case report and literature review

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Perivascular epithelioid cell tumor of the liver complicated with anemia: a case report and literature review