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Secondary deep vein thrombosis associated with iliac lymph node metastasis of an unknown primary tumor has not been previously reported. The patient was a 57-year-old male with persistent right leg edema. Computed tomography demonstrated a mass surrounding the right external iliac vessels, and deep vein thrombosis in the right external iliac and femoral veins. Physical, laboratory, and imaging examinations did not reveal any further tumor. The patient was diagnosed with deep vein thrombosis associated with right iliac lymph node metastasis of an unknown primary tumor. Complete resection of the tumor along with the involved vessels and vascular reconstruction was performed.
Cancer of unknown primary (CUP) has been mostly described in terms of cervical, axillary, or inguinal lymph node metastases, and several authors have made recommendations regarding diagnosis, treatment, and follow-up. However, secondary deep vein thrombosis associated with iliac lymph node metastasis of CUP has not previously been reported.
We herein describe a rare case of deep vein thrombosis due to iliac lymph node metastasis of CUP in a 57-year-old male. Complete surgical resection and vascular reconstruction eliminated symptoms.
A 57-year-old previously healthy male with persistent right leg edema was referred to our institution. His right leg was swollen without spontaneous pain or tenderness. The right dorsal artery, popliteal artery and femoral artery were well palpated, similarly to the left-sided vessels. The inguinal and cervical lymph nodes were not palpated. Computed tomography (CT) showed a 53 × 44 mm heterogeneously enhanced tumor in the right retroperitoneal space in the pelvis, which involved the right external iliac artery and vein, and deep vein thrombosis (DVT) in the right external iliac and femoral veins (Fig. 1A and 1B1B). His laboratory data showed normal findings. Assays for antithrombin, protein C activity, protein S antigen, lupus anticoagulant, anticardiolipin IgG, prothrombin time, activated partial thromboplastin time, fibrin and fibrinogen degradation products and D-dimer were performed. Only D-dimer (1.3 mg/ml) deviated slightly from the normal range (normal values: ≤1.0 mg/ml). Urinalysis and stool occult blood testing was negative. Before surgery, the squamous cell carcinoma antigen (SCC) level was elevated to 3.5 ng/ml (normal values: 0–1.5 ng/ml), while carcinoembryonic antigen (CEA), carbohydrate antigen 19-9 (CA19-9), and CA125 were all within the normal ranges. Although metastatic lymph node was suspected, no primary lesions were detected by systemic CT, abdominal and pelvic magnetic resonance imaging (MRI), esophagogastroduodenoscopy or colonoscopy. The patient was diagnosed with DVT associated with right iliac lymph node metastasis of an unknown primary tumor.
Extirpation of the tumor was performed to relieve the symptoms and make a histopathological diagnosis. The tumor was 50 mm in diameter and involving the right external iliac artery and vein (Fig. 2A). It was resected en bloc with part of the external iliac artery and vein (Fig. 2B). Resection margins were clear of tumor. The external artery was reconstructed with an 8 mm expanded polytetrafluoroethylene (ePTFE) graft. After thrombectomy using a 4 French Fogarty catheter, the external iliac vein was reconstructed with a left femoral great saphenous vein graft (Fig. 2C). Histological examination of the mass revealed a proliferation of squamoid cancer cells forming intercellular bridges and a solid nest with extensive coagulative necrosis in the lymph nodes (Fig. 3). The definitive pathological diagnosis was metastatic squamous cell carcinoma. To maintain graft patency, the patient was initially treated immediately after surgery with intravenous heparin, which was continued until warfarin was fully effective, with a target international normalized ratio of between 2.5 and 3.0. Additionally, 100 mg/day aspirin was administered.
A CT scan of the whole body was performed again 7 days after surgery; however, primary lesions were not detected. Moreover, positron emission tomography (PET) CT and MRI were performed 20 days after surgery. However, fluorodeoxy glucose (FDG) accumulation was recognized only at the site of surgery, and primary lesions were not detected.
Postoperative radiation therapy and chemotherapy were not performed because the patient refused them. A systemic physical examination, SCC of serum tumor marker, chest radiograph, CT of the abdomen and pelvis have been performed 6 months after surgery, and the patient experienced no local recurrence or primary cancer and reconstructed external artery and vein were patent.
Cancer of unknown primary (CUP) is defined as histologically confirmed metastatic cancer in which a complete physical examination, full blood count and biochemistry, urinalysis and stool occult blood testing, chest radiography, and CT of the abdomen and pelvis fail to identify the primary site at the time of first diagnosis, regardless of the follow-up duration.1,2) Cancer of unknown primary accounts for 3%–5% of all malignancies, and 15% of these represent squamous cell carcinoma (SCC).2) Squamous cell carcinoma of CUP is usually detected as cervical or inguinal lymph node metastasis. To our knowledge, there has been only one report of a case of CUP involving iliac lymph node metastasis.3)
Treatment for CUP remains controversial. Therapy should be tailored on an individual basis by recognition of well-defined clinicopathologic subsets that differ in prognosis.2) Locoregional management by lymph node complete resection is recommended for patients with SCC of CUP.4) The role of radiation therapy and chemotherapy is not established. In our case, the tumor was resected completely and the patient refused strongly postoperative radiation therapy and chemotherapy. Postoperative radiation therapy and chemotherapy were not performed.
Because of the relatively poor long-term patency, venous reconstruction has not been as successful as arterial reconstruction and there are no criteria regarding which type of material is best for vein replacement. Current published data suggest high rates of PTFE graft occlusion in a series of cases, with a patency of 62% at 3 years reported by Jost and 60% at 12 months by Caldarelli.5,6) At the same time, patency of an autologous vein graft appears to be higher.5) In our case, we used a saphenous vein graft and the patient has been receiving anticoagulation therapy during his lifetime to improve patency.
In this report, we presented a case of DVT associated with CUP with lymph node metastasis localized around the right iliac vessels. Complete surgical resection and vascular reconstruction eliminated symptoms.
The authors have no conflicts of interest to declare.