Department of Clinical Sciences of Companion Animals, Faculty of Veterinary Medicine, Utrecht University, Yalelaan 108, 3584 CM, Utrecht, The Netherlands.
Cytopath Veterinary Pathology, PO Box 24, Ledbury, Herefordshire, HR8 2YD, UK.
Acta Vet Scand. 2020 Feb 1;62(1):8. doi: 10.1186/s13028-020-0506-9.
Lymphatic vascular malformations are rare findings in canine patients with six reports available in veterinary literature. Retroperitoneal cystic lymphatic malformations have not been described previously in canine patients and neither has the use of immunohistochemistry to determine their origin, i.e. vascular versus lymphatic.
An 8-year-old neutered female Cocker spaniel was referred for pollakiuria, dysuria and a painful abdomen. Computed tomography scanning of the abdomen showed a fluid filled structure adjacent to the urinary bladder. During surgical exploration, a thin walled cystic structure with sero-haemorrhagic fluid was found, extending from the retroperitoneal space into the abdomen. The mass was excised and submitted for histopathology, revealing a cystic mass lined by a fibrovascular capsule within the retroperitoneal/mesenteric adipose tissue. The inner surface of the cyst was lined by a single layer of bland, flattened spindle cells. Intramural blood vessels were well differentiated, with perivascular haemorrhage. On recurrence 11 months later, the mass was excised for the second time and a PleuralPort (Norfolk Animal products) was placed. Fifteen months after initial presentation, progression occurred with haemorrhagic fluid in the cystic space, pleural- and abdominal cavities and the owners opted for euthanasia. Histopathology and positive immunohistochemistry for lymphatic markers lymphatic vessel endothelial hyaluronic acid receptor-1 (LYVE-1) and prospero homeobox protein-1 (PROX-1) confirmed a lymphatic vascular origin of the cystic structure.
To our experience, a definitive diagnosis of retroperitoneal cystic malformation of lymphatic origin could be done only by combining the clinical presentation, advanced imaging, histopathology and LYVE-1 and PROX-1 immunohistochemistry. This is the first report of a vascular malformation in a dog where immunohistochemistry was used to make a final diagnosis. A lymphatic malformation, even if rare, should be added on the list of the differential diagnosis in a patient with a retroperitoneal cystic structure containing serohaemorrhagic fluid. Results of this case report can aid in diagnosis of future cases, however, further studies on therapy and management are needed to provide additional information about optimal treatment of these patients.
犬淋巴管畸形较为罕见,兽医文献中仅有 6 篇相关报道。犬腹膜后囊性淋巴管畸形此前尚未见报道,也未见使用免疫组织化学来确定其起源,即血管起源还是淋巴管起源。
一只 8 岁已绝育雌性可卡犬因多尿、尿痛和腹痛就诊。腹部计算机断层扫描显示膀胱旁有一个充满液体的结构。在手术探查过程中,发现一个薄壁囊性结构,内含浆液血性液体,从腹膜后间隙延伸至腹部。切除该肿块并送检组织病理学检查,结果显示在腹膜后/肠系膜脂肪组织内,有一个囊性肿块,由纤维血管囊包绕。囊肿内表面由单层平坦的梭形细胞衬覆。壁内血管分化良好,伴血管周围出血。11 个月后复发时,第二次切除肿块并放置了胸膜Port(Norfolk Animal products)。初次就诊 15 个月后,肿块再次进展,囊性腔内出现血性液体,胸腔和腹腔也有血性液体,主人选择对犬实施安乐死。组织病理学和淋巴管内皮透明质酸受体 1(LYVE-1)和 Prospero 同源框蛋白 1(PROX-1)的免疫组织化学阳性结果证实了囊性结构的淋巴管血管起源。
根据我们的经验,只有将临床表现、先进的影像学检查、组织病理学和 LYVE-1 和 PROX-1 免疫组织化学相结合,才能对腹膜后囊性淋巴管畸形做出明确诊断。这是首例使用免疫组织化学做出最终诊断的犬血管畸形病例。即使罕见,对于含有浆液血性液体的腹膜后囊性结构的患者,也应将淋巴管畸形纳入鉴别诊断。本病例报告的结果有助于未来病例的诊断,但需要进一步研究治疗和管理,以提供关于这些患者最佳治疗的更多信息。