Oligometastasis to the testes from oesophageal adenocarcinoma

Background

9100 people are diagnosed with oesophageal cancer each year in the United Kingdom and is the sixth leading cause of cancer mortality worldwide.1 Patients who are deemed suitable for radical treatment after multimodal staging are offered either immediate surgery or neoadjuvant treatment followed by surgery depending on the stage of the disease.2 About 40% of patients are suitable for such radical treatment.3 Those with a stage IIA or above disease is offered either neoadjuvant chemotherapy (in the form of epirubicin/cisplatin/5-fluorouracil (ECF) or 5-fluorouracil/leucovorin/oxplatin/docetaxel (FLOT))4 or chemoradiation in the form of the chemoradiotherapy for oesophageal cancer followed by surgery study (CROSS) protocol)5 prior to resection. Although, some patients have a complete pathological response to treatment from these neoadjuvant treatments, and while this might confer some survival benefit of up to a 30% increase in 3 year survival, there is no reliable way to identify the level of tumour regression apart from oesophagectomy.6–8 The common metastatic sites for oesophageal adenocarcinoma are liver, distant lymph nodes, lungs, bone and brain.9 Metastases to the female reproductive system from upper gastrointestinal tract cancers are well documented and unfortunately carry a poor prognosis.10 Metastases to the testes on the other hand are unusual and there has only been two cases of adenocarcinoma from the oesophagus.11 12

Case presentation

A man in his early 70s presented to our Regional Oesophagogastric Cancer Unit with a Siewart 1 gastroesophageal junctional cancer. He had a history of hypertension and had previously undergone a left inguinal hernia repair. His WHO performance status was 0. Pathology from endoscopic biopsies confirmed this to be a poorly differentiated adenocarcinoma. He underwent multimodal staging according to our Upper Gastrointestinal (UGI) Multidisciplinary Team (MDT) protocol with CT, fluorodeoxyglucose positron emission tomography (FDG-PET) and endoscopy. An incidental small bowel lesion was also identified which was consistent with a gastrointestinal stromal tumour (GIST) (figure 1). FDG-PET showed an avid primary oesophagogastric junctional (OGJ) tumour (maximum Standard Unit Value (SUVmax) of 11.1) and also identified an avid left supraclavicular node (figure 2). This was further delineated by ultrasound (figure 3) and fine-needle aspiration, which confirmed the node to be benign, with the histology showing only increased lymphocytes with no concerning features or any sign of malignancy. Similarly, an area of FDG uptake correlating to the top of segment 8 on the liver was further investigated with an MRI. This confirmed the liver to be normal, with the area of FDG uptake to correspond to focal consolidation in the lung which was superimposed on the FDG-PET images due to respiration artefact.

Figure 1

Gastrointestinal stromal tumour on CT.

Figure 2

Supraclavicular node hot spot.

Figure 3

Neck ultrasound demonstrating the lymph node demonstrated on PET.

All investigations and results were analysed and discussed at our MDT and he was staged as T3N0M0 according to TNM 8 classification.13

Following discussion at the MDT, he was advised to undergo chemoradiation followed by oesophagogastrectomy to which he agreed after consultation in the outpatient department. There were no significant adverse events during neoadjuvant chemoradiotherapy (according to the CROSS protocol) and, 2 weeks following completion, a further FDG-PET scan was organised. This demonstrated a significant reduction in SUV max of the OGJ tumour from 11.1 to 7.3 and there was no evidence of any new lesions, metastatic disease or unusual uptake in the groin region (figure 4A,B).

Figure 4

Preoperative FDG-PET showing (A) avid oesophageal tumour and (B) no abnormal uptake in the groin and genitalia.

A two-stage Ivor-Lewis oesophagogastrectomy was performed with a simultaneous small bowel resection of the presumed GIST which was found on staging. All specimens were sent to pathology for further examination. He had an inpatient stay of 17 days which was complicated by delayed gastric emptying and had an endoscopic injection of 100 units botulinum toxin A into his pylorus which resolved his symptoms prior to discharge.

His histology showed a complete response to treatment (Mandard grade 1). There was a tiny focus of high-grade glandular dysplasia in a segment of barrett’s which was completely excised with the specimen. 23 lymph nodes were excised with the specimen, of which none were involved. The final staging was ypT0N0 R0 according to TNM 8.

The suspected small bowel GIST was found to be a low-risk spindle cell-GIST with 1–2 mitosis per 5 mm² and the typical features with immunohistochemistry showing immunopositivity for CD117, DOG1, (patchy) CD34 and (patchy weak) SMA. The tumour was immunonegative for S100, desmin and MNF116. This completely excised and was staged at T2N0 R0 according to TNM 8.

He was reviewed in the outpatient department 2 weeks after discharge and a further botulinum toxin injection was planned for symptoms in 6 weeks.

Four months later he presented to the urologists after urgent referral from the GP with a right testicular swelling. An ultrasound scan of the testes was organised which demonstrated a focal mass (3.3 cm x 2.2 cm x 3.8 cm) with vascularity in the right testes in keeping with possible malignancy (figure 5). A CT scan did not demonstrate any other sites of disease and he went on to have a radical right orchidectomy via the groin due to a suspicion of a primary testicular malignancy.

Figure 5

Right testicular ultrasound with Doppler.

Outcome and follow-up

The postoperative histology from his right testes found poorly differentiated carcinoma infiltrating into the testes. Markers of primary testicular tumours and tumours which metastasise commonly to the testes such as germ cell tumours (OCT 3–4), lymphomas (LCA) and melanomas (S100) were negative. Further immunohistochemistry was done which was positive for CK7 and negative for CK20, CK 5/6 and p63. The morphology of the tumour was felt to be identical to the histology from the oesophagus taken preoperatively by the reporting pathologist. Thus, the tumour was deemed to be a metastatic deposit from the patient’s previous known oesophageal cancer.

He was discussed at both the urology and upper gastrointestinal MDT. The consensus of both MDTs was that the testicular tumour was a secondary from his primary OGJ tumour, and as there were no distant metastases found no further treatment was required.

However, a few weeks later, he was seen in the UGI clinic with back pain and weight loss. A PET scan was arranged at this point, which found significant retroperitoneal lymphadenopathy. After further discussion at the MDT, he was referred to clinical oncology for palliative radiotherapy.