AFP-producing adenocarcinoma of the esophagogastric junction: report of a case with atypical immunohistochemical findings responding to palliative chemotherapy with 5-fluorouracil, leucovorin, oxaliplatin, and docetaxel (FLOT regime)

Abstract

AFP-producing adenocarcinoma of the esophagus and esophagogastric junction are rare tumor diseases. These tumors show an aggressive behavior characterized by early occurrence of liver metastases and mimic hepatocellular carcinoma (HCC). A general recommendation for palliative therapy is not established for these special tumors.

Here we report about a 61-year-old man with multiple liver metastases and high serum alpha-fetoprotein (AFP) level. First, HCC was suspected, but further evaluation showed an AFP-producing adenocarcinoma of the esophagogastric junction with unusual findings on further immunohistochemical analysis. Palliative chemotherapy with FLOT (5-fluorouracil, leucovorin, oxaliplatin, and docetaxel) regime showed a 9 month duration of partial response.

Zusammenfassung

AFP-produzierende Adenokarzinome des Ösophagus und ösophago-gastralen Übergangs sind seltene Tumorerkrankungen. Diese Tumore zeigen ein aggressives Wachstumsverhalten und sind gekennzeichnet durch eine frühe hepatische Metastasierung. Histopathologisch gibt es Ähnlichkeiten zum hepatozellulären Karzinom (HCC). Die palliative Therapie dieser Tumoren ist bislang nicht standardisiert.

Wir berichten über einen 61-jährigen Patienten mit multiplen Lebermetastasen und einer hohen Serum-Alpha-Fetoprotein- (AFP) Konzentration. Zunächst wurde ein HCC vermutet, jedoch erbrachten die weiteren Untersuchungen den Nachweis eines AFP-produzierenden Adenokarzinoms des ösophago-gastralen Übergangs mit ungewöhnlichen Befunden in der immunhistochemischen Analyse. Unter einer palliativen Chemotherapie nach dem FLOT-Regime (5-Fluorouracil, Leucovorin, Oxaliplatin and Docetaxel) zeigte sich eine partielle Tumorremission für eine Zeitdauer von neun Monaten.

Schlüsselwörter

Ösophaguskarzinom - Magenkarzinom - Barrett-Oesophagus

Key words

esophagus carcinoma - gastric cancer - barrett esophagus

• References

• 1 Sawada H, Watanabe A, Yamada Y et al. Alpha-fetoprotein-producing esophageal adenocarcinoma: report of a case. Surg Today 1993; 23: 1103-1107
• 2 Kashani A, Ellis JC, Kahn M et al. Liver metastasis from hepatoid adenocarcinoma of the esophagus mimicking hepatocellular carcinoma. Gastroenterol Rep (Oxf) 2015; [Epub ahead of print]
• 3 Kripp M, Ströbel P, Dinter D et al. Alpha-fetoprotein expressing metastastic adenocarcinoma of the esophago-gastric junction responding favorably to capecitabine and oxaliplatin. Anticancer Drugs 2009; 20: 75-78
• 4 Chiba N, Yoshioka T, Sakayori M et al. AFP-producing hepatoid adenocarcinoma in association with Barrett's esophagus with multiple liver metastasis responding to paclitaxel/CDDP: a case report. Anticancer Res 2005; 25: 2965-2968
• 5 Shimakawa T, Ogawa K, Naritaka Y et al. Alpha-fetoprotein producing Barrett’s esophageal adenocarcinoma: a case report. Anticancer Res 1999; 19: 4369-4373
• 6 Tanigawa H, Kida Y, Kuwao S et al. Hepatoid adenocarcinoma in Barrett’s esophagus associated with achalasia: first case report. Pathol Int 2002; 52: 141-146
• 7 Motoyama T, Higuchi M, Taguchi J. Combined choriocarcinoma, hepatoid adenocarcinoma, small cell carcinoma and tubular adenocarcinoma in the oesophagus. Virchows Arch 1995; 427: 451-454
• 8 Kuroda N, Onishi K, Lee GH. Combined tubular adenocarcinoma and hepatoid adenocarcinoma arising in Barrett esophagus. Ann Diagn Pathol 2011; 15: 450-453
• 9 Atiq M, Husain M, Dang S et al. Hepatoid esophageal carcinoma: a rare cause of elevated alpha fetoprotein. J Gastrointest Cancer 2008; 39: 58-60
• 10 Kobayashi N, Ohbu M, Kuroyama S et al. Alpha-fetoprotein-producing esophageal adenocarcinoma: report of a case. Surg Today 2001; 31: 915-919
• 11 Kawai H, Sekine S, Sanada T et al. Alpha-fetoprotein-producing esophageal carcinoma: a case report. Anticancer Res 2003; 23: 3837-3840
• 12 Chang YC, Nagasue N, Abe S et al. Comparison between the clinicopathologic features of AFP-positive and AFP-negative gastric cancers. Am J Gastroenterol 1992; 87: 321-325
• 13 Chang YC, Nagasue N, Kohno H et al. Clinicopathologic features and long-term results of alpha-fetoprotein-producing gastric cancer. Am J Gastroenterol 1990; 85: 1480-1485
• 14 Koide N, Nishio A, Igarashi J et al. Alpha-fetoprotein-producing gastric cancer: histochemical analysis of cell proliferation, apoptosis, and angiogenesis. Am J Gastroenterol 1999; 94: 1658-1663
• 15 Kamoshida S, Suzuki M, Sakurai Y et al. Expression of chemoresistance-related proteins in alpha-fetoprotein-producing adenocarcinoma of the digestive organs. Oncol Rep 2006; 16: 721-727
• 16 Wahren B, Harmenberg J, Edsmyr F et al. Possible tumour markers in patients with oesophagus cancer. Scand J Gastroenterol 1979; 14: 361-365
• 17 Cao D, Humphrey PA, Allan RW. SALL4 is a novel sensitive and specific marker for metastatic germ cell tumors, with particular utility in detection of metastatic yolk sac tumors. Cancer 2009; 115: 2640-2651
• 18 Ushiku T, Shinozaki A, Shibahara J et al. SALL4 represents fetal gut differentiation of gastric cancer, and is diagnostically useful in distinguishing hepatoid gastric carcinoma from hepatocellular carcinoma. Am J Surg Pathol 2010; 34: 533-540
• 19 Yong KJ, Chai L, Tenen DG. Oncofetal gene SALL4 in aggressive hepatocellular carcinoma. N Engl J Med 2013; 369: 1171-1172
• 20 Al-Batran SE, Hartmann JT, Hofheinz R et al. Biweekly fluorouracil, leucovorin, oxaliplatin, and docetaxel (FLOT) for patients with metastatic adenocarcinoma of the stomach or esophagogastric junction: a phase II trial of the Arbeitsgemeinschaft Internistische Onkologie. Ann Oncol 2008; 19: 1882-1887
• 21 Schulz C, Kullmann F, Kunzmann V et al. NeoFLOT: Multicenter phase II study of perioperative chemotherapy in resectable adenocarcinoma of the gastroesophageal junction or gastric adenocarcinoma-Very good response predominantly in patients with intestinal type tumors. Int J Cancer 2015; 137: 678-685
• 22 Anter AH, Abdel-Latif RM. The safety and efficacy of fluorouracil, leucovorin, oxaliplatin, and docetaxel (FLOT) combination in the front-line treatment for patients with advanced gastric or gastroesophageal adenocarcinoma: phase II trial. Med Oncol 2013; 30: 451