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一名青少年男性的肠系膜囊性淋巴管瘤;诊断难题:病例报告

Mesenteric cystic lymphangioma in an adolescent male; a diagnostic dilemma: A case report.

作者信息

Bhusal Asmita, Habibullah Quazi, Rahman Mashiur, Bandh Biplob, Islam Saiful, Dola Tanvin, Bista Saugat

机构信息

Sir Salimullah Medical College and Mitford Hospital, Dhaka, Bangladesh.

Department of Surgery, Sir Salimullah Medical College and Mitford Hospital, Dhaka, Bangladesh.

出版信息

Int J Surg Case Rep. 2023 Dec;113:109042. doi: 10.1016/j.ijscr.2023.109042. Epub 2023 Nov 14.

Abstract

INTRODUCTION AND IMPORTANCE

Hemolymphangioma, a rare type of lymphangioma, is a benign hamartoma of the blood vessels and lymphatic system. Considered to be extremely rare among adults with infrequent occurrence in abdominal regions, lymphangioma involving mesentery accounts for <1 % of cases and only 0.05 % involve the GI tract. Due to the absence of typical clinical presentation, making a confirmatory preoperative diagnosis is challenging.

CASE PRESENTATION

An 18-year-old Bangladeshi man visited the hospital with the complaints of epigastric and umbilical pain for 15 days which was insidious in onset. Physical examination revealed an ill defined lump that was palpable on the right side of the abdomen. Ultrasonography showed multiloculated cystic mass having septation approximately 13 × 6 cm in size. An abdominal CT scan showed cystic lesion with septations measuring about 14.5 × 12.3 cm, compressing the bowel loops towards left, that was suggestive of mesenteric lymphangioma. The patient underwent partial surgical resection. The excised mass was sent for histopathology. Histopathology disclosed a benign tumor composed of proliferation of blood vessels of different sizes lined by endothelium in a fibromuscular stroma.

CLINICAL DISCUSSION

Mesenteric lymphangiomas are infrequent malformations and very few cases of mesenteric hemolymphangioma have been reported in adults. Histopathology is required for confirmatory diagnosis and immunohistochemistry is required to differentiate the tumor subtype. Surgical resection is deemed a standard treatment modality.

CONCLUSION

We report an extremely rare case of mesenteric hemolymphangioma to bring it to concern that even with the vague clinical presentations and involvement of age groups beyond the status quo, surgeons must be vigilant about abdominal MCL/hemolymphangioma and proceed accordingly.

摘要

引言与重要性

血淋巴管瘤是一种罕见的淋巴管瘤类型,是血管和淋巴系统的良性错构瘤。肠系膜淋巴管瘤在成年人中极为罕见,在腹部区域很少发生,占病例的比例不到1%,仅有0.05%累及胃肠道。由于缺乏典型的临床表现,术前做出确诊诊断具有挑战性。

病例介绍

一名18岁的孟加拉国男子因上腹部和脐周疼痛15天前来就诊,疼痛起病隐匿。体格检查发现腹部右侧可触及一个边界不清的肿块。超声检查显示一个多房性囊性肿块,大小约为13×6厘米,有分隔。腹部CT扫描显示一个有分隔的囊性病变,大小约为14.5×12.3厘米,将肠袢向左挤压,提示肠系膜淋巴管瘤。患者接受了部分手术切除。切除的肿块送去做组织病理学检查。组织病理学显示为一个良性肿瘤,由不同大小的血管在内皮细胞衬里的纤维肌性基质中增生组成。

临床讨论

肠系膜淋巴管瘤是罕见的畸形,成人肠系膜血淋巴管瘤的病例报道极少。确诊需要组织病理学检查,鉴别肿瘤亚型需要免疫组织化学检查。手术切除被认为是标准的治疗方式。

结论

我们报告了一例极其罕见的肠系膜血淋巴管瘤病例,以引起关注,即即使临床表现模糊且涉及现有年龄组之外的人群,外科医生也必须警惕腹部肠系膜囊肿性淋巴管瘤/血淋巴管瘤并相应地进行处理。

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