AccScience Publishing / TD / Online First / DOI: 10.36922/td.3736
CASE REPORT

Signet ring cell carcinoma of the urinary bladder presenting with carcinocythemia and skeletal metastasis: A case report

Emma Rullo1* Piergianni Biondi2 Chiara Nicolazzo3 Walter Gianni2 Luciano Colangelo2 Mirella Cilli2 Paola Gazzaniga3 Mara Riminucci1 Alessandro Corsi1 Salvatore Minisola2
Show Less
1 Department of Molecular Medicine, Sapienza University, and Section of Pathology, Policlinico Umberto I, Rome, Italy
2 Department of Clinical, Internal, Anesthesiologic and Cardiovascular Sciences, Sapienza University, and Mineral Metabolism Section, Policlinico Umberto I, Rome, Italy
3 Department of Molecular Medicine, Sapienza University, and Cancer Liquid Biopsy Unit, Policlinico Umberto I, Rome, Italy
Tumor Discovery, 3736 https://doi.org/10.36922/td.3736
Submitted: 22 May 2024 | Accepted: 7 August 2024 | Published: 8 October 2024
© 2024 by the Author(s). This article is an open access article distributed under the terms and conditions of the Creative Commons Attribution 4.0 International License ( https://creativecommons.org/licenses/by/4.0/ )
Abstract

Cancer of unknown primary accounts for approximately 3 – 5% of all malignancies and is typically associated with a dismal prognosis. We describe a 65-year-old man who presented with skeletal metastasis and circulating tumor cells exhibiting signet ring (SR) morphology. The patient was diagnosed with SR cell carcinoma (SRCC) through a bone marrow biopsy. This case report aimed to emphasize the importance of clinicians’ awareness of SRCC of the urinary bladder. The primary site of tumor origin was not identified as antemortem. The patient died 2 months after being admitted for pulmonary embolism. At autopsy, the urinary bladder was determined to be the primary site of the tumor. Primary SRCC of the urinary bladder is extremely rare. There are currently no established consensus guidelines for its management. Surgery continues to be the primary treatment option when the condition is localized.

Keywords
Cancer of unknown primary
Signet ring cell carcinoma
Urinary bladder carcinoma
Carcinocythemia
Funding
None.
Conflict of interest
The authors declare that they have no competing interests.
References
  1. Krämer A, Bochtler T, Pauli C, et al. Cancer of unknown primary: ESMO Clinical Practice Guideline for diagnosis, treatment and follow-up. Ann Oncol. 2023;34(3):228-246. doi: 10.1016/j.annonc.2022.11.013

 

  1. Levi F, Te VC, Erler G, Randimbison L, La Vecchia C. Epidemiology of unknown primary tumours. Eur J Cancer. 2002;38(13):1810-1812. doi: 10.1016/s0959-8049(02)00135-1

 

  1. Massard C, Loriot Y, Fizazi K. Carcinomas of an unknown primary origin--diagnosis and treatment. Nat Rev Clin Oncol. 2011;8(12):701-710. doi: 10.1038/nrclinonc.2011.158

 

  1. Pavlidis N, Fizazi K. Carcinoma of unknown primary (CUP). Crit Rev Oncol Hematol. 2009;69(3):271-278. doi: 10.1016/j.critrevonc.2008.09.005

 

  1. Oien KA. Pathologic evaluation of unknown primary cancer. Semin Oncol. 2009;36(1):8-37. doi: 10.1053/j.seminoncol.2008.10.009

 

  1. Beauchamp K, Moran B, O’Brien T, et al. Carcinoma of unknown primary (CUP): An update for histopathologists. Cancer Metastasis Rev. 2023;42:1189-1200. doi: 10.1007/s10555-023-10101-6

 

  1. Celik O, Budak S, Ekin G, Akarken I, Ilbey YO. A case with primary signet ring cell adenocarcinoma of the prostate and review of the literature. Arch Ital Urol Androl. 2014;86(2):148-149. doi: 10.4081/aiua.2014.2.148

 

  1. Lendorf ME, Dohn LH, Dunga BA, Loya AC, Pappot H. An updated review on primary signet-ring cell carcinoma of the urinary bladder and report of a case. Scand J Urol. 2018;52(2):87-93. doi: 10.1080/21681805.2017.1418020

 

  1. Benerjee N, Parmar K, Vaiphei K. Primary signet-ring cell carcinoma of the urinary bladder. Autops Case Rep. 2021;11:e2021264. doi: 10.4322/acr.2021.264

 

  1. Ivanov A, Antonov P, Zapryanov M, Uchikov P, Belovezhdov V. Primary signet-ring cell adenocarcinoma of the bladder-A case report and review of literature. Urol Case Rep. 2022;42:102022. doi: 10.1016/j.eucr.2022.102022

 

  1. Bagaporo Larrazabal R Jr., Cheng PVC, David-Wang A, Requiso D. Signet-ring cell adenocarcinoma of unknown primary presenting with superior vena cava (SVC) syndrome: Rare type of cancer. BMJ Case Rep. 2019;12(12):e232269. doi: 10.1136/bcr-2019-232269

 

  1. Nicolazzo C, Raimondi C, Gradilone A, et al. Circulating tumor cells in right- and left-sided colorectal cancer. Cancers (Basel). 2019;11(8):1042. doi: 10.3390/cancers11081042

 

  1. DeMay RM, Grathwohl MA. Signet-ring-cell (colloid) carcinoma of the urinary bladder. Cytologic, histologic and ultrastructural findings in one case. Acta Cytol. 1985;29(2):132-136.

 

  1. Carey RW, Taft PD, Bennett JM, Kaufman S. Carcinocythemia (carcinoma cell leukemia). An acute leukemia-like picture due to metastatic carcinoma cells. Am J Med. 1976;60(2):273-278. doi: 10.1016/0002-9343(76)90437-x

 

  1. Ronen S, Kroft SH, Olteanu H, Hosking PR, Harrington AM. Carcinocythemia: A rare entity becoming more common? A 3-year, single institution series of seven cases and literature review. Int J Lab Hematol. 2019;41(1):69-79. doi: 10.1111/ijlh.12924

 

  1. Krawczyk P, Jassem J, Wojas-Krawczyk K, Krzakowski M, Dziadziuszko R, Olszewski W. New genetic technologies in diagnosis and treatment of cancer of unknown primary. Cancers (Basel). 2022;14(14):3429. doi: 10.3390/cancers14143429

 

  1. Hayashi H, Takiguchi Y, Minami H, et al. Site-specific and targeted therapy based on molecular profiling by next-generation sequencing for cancer of unknown primary site: A nonrandomized phase 2 clinical trial. JAMA Oncol. 2020;6(12):1931-1938. doi: 10.1001/jamaoncol.2020.4643

 

  1. Lu MY, Chen TY, Williamson DFK, et al. AI-based pathology predicts origins for cancers of unknown primary. Nature. 2021;594(7861):106-110. doi: 10.1038/s41586-021-03512-4
Share
Back to top
Tumor Discovery, Electronic ISSN: 2810-9775 Print ISSN: 3060-8597, Published by AccScience Publishing