Ohkura Y, Uruga H, Shiiba M, Ito S, Shimoyama H, Ishihara M, Ueno M, Udagawa H. Phosphoglyceride crystal deposition disease requiring differential diagnosis from malignant tumors and confirmed by Raman spectroscopy: A case report. World J Clin Cases 2022; 10(23): 8304-8311 [PMID: 36159533 DOI: 10.12998/wjcc.v10.i23.8304]
Corresponding Author of This Article
Yu Ohkura, MD, PhD, Assistant Professor, Department of Gastroenterological Surgery, Toranomon Hospital, 2-2-2 Toranomon, Minato-ku, Tokyo 105-8470, Japan. jag.ptj69-or.jpajhq.107emtg@docomo.ne.jp
Research Domain of This Article
Gastroenterology & Hepatology
Article-Type of This Article
Case Report
Open-Access Policy of This Article
This article is an open-access article which was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution Non Commercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: http://creativecommons.org/licenses/by-nc/4.0/
Yu Ohkura, Hayato Shimoyama, Masaki Ueno, Harushi Udagawa, Department of Gastroenterological Surgery, Toranomon Hospital, Tokyo 105-8470, Japan
Hironori Uruga, Shinji Ito, Department of Diagnostic Pathology, Toranomon Hospital, Tokyo 105-8470, Japan
Masato Shiiba, Makiko Ishihara, Department of Diagnostic Imaging Center, Toranomon Hospital, Tokyo 105-8470, Japan
Author contributions: Ohkura Y, Uruga H and Ito S designed and conducted the research and wrote the paper; Ohkura Y, Udagawa H and Ito S drafted the article, revised it critically for important intellectual content, and gave final approval for the content; Ohkura Y, Uruga H, Shiiba M, Ito S, Shimoyama H, Ishihara M, Ueno M and Udagawa H created study materials or recruited patients; all authors read and approved the final manuscript.
Informed consent statement: The patient provided written informed consent for publication of this case report and the associated images.
Conflict-of-interest statement: All the authors report no relevant conflicts of interest for this article.
CARE Checklist (2016) statement: The authors have read the CARE Checklist (2016), and the manuscript was prepared and revised according to the CARE Checklist (2016).
Open-Access: This article is an open-access article that was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution NonCommercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: https://creativecommons.org/Licenses/by-nc/4.0/
Corresponding author: Yu Ohkura, MD, PhD, Assistant Professor, Department of Gastroenterological Surgery, Toranomon Hospital, 2-2-2 Toranomon, Minato-ku, Tokyo 105-8470, Japan. jag.ptj69-or.jpajhq.107emtg@docomo.ne.jp
Received: February 7, 2022 Peer-review started: February 7, 2022 First decision: April 10, 2022 Revised: April 11, 2022 Accepted: June 30, 2022 Article in press: June 30, 2022 Published online: August 16, 2022 Processing time: 175 Days and 6 Hours
Abstract
BACKGROUND
Phosphoglyceride crystal deposition disease (PCDD) is a rare acquired disease in which phospholipid crystals deposit in bone and soft tissue long after surgery, trauma, or repeated injections.
CASE SUMMARY
A 60-year-old-woman was referred to our department because of multiple abdominal masses after open splenectomy for idiopathic thrombocytopenic purpura 29 years earlier. All the masses showed marked fluorodeoxyglucose (FDG) uptake on 18F-fluorodeoxyglucose positron emission tomography (18F-FDG-PET) and were strongly suspected to be malignant tumors. Surgical biopsies were performed, and the abdominal masses were found to be aligned vertically, three in a row, along the tissue layers cut in the patient’s previous surgery. Pathological finding of the specimens showed foreign body granuloma consisting of histiocytes and multinucleated giant cells accumulating around needle-like crystals. The crystals were confirmed as phosphoglyceride by Raman spectroscopy, and PCDD was diagnosed. To our knowledge, this is the first report of PCDD diagnosed by Raman spectroscopy.
CONCLUSION
We made a definitive diagnosis of PCDD in a patient with multiple tumors showing marked FDG uptake on 18F-FDG-PET by incisional biopsy and compo- sition analysis using Raman spectroscopy, a method that has not previously been reported for the diagnosis of PCDD.
Core Tip: Here, we report a case in which Raman spectroscopy proved useful for differential diagnosis of phosphoglyceride crystal deposition disease (PCDD) in a patient with multiple malignant abdominal masses showing extremely high uptake on 18F-fluorodeoxyglucose positron emission tomography, many years after undergoing open splenectomy. Initially, we strongly suspected the masses to be malignant. To rule out malignancy, we performed surgical biopsies and the pathological diagnosis was foreign body granulomas with crystal deposition. Raman spectroscopy led to the definitive diagnosis of PCCD. Raman spectroscopy is a simple and precise method for diagnosing PCCD.
