{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,2,24]],"date-time":"2026-02-24T10:18:28Z","timestamp":1771928308389,"version":"3.50.1"},"reference-count":37,"publisher":"MDPI AG","issue":"4","license":[{"start":{"date-parts":[[2026,2,13]],"date-time":"2026-02-13T00:00:00Z","timestamp":1770940800000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Diagnostics"],"abstract":"<jats:p>Background and Clinical Significance: Phosphoglyceride crystal deposition disease (PCDD) is an extremely rare condition characterized by the deposition of phosphoglyceride crystals, occasionally forming tumor like lesions that present significant diagnostic challenges. Here, we report, to our knowledge, the first documented recurrent case of PCDD confined to the mandible, which clinically and radiologically mimicked a malignant bone tumor. Case Presentation: An 80-year-old female patient presented with a progressively enlarging mandibular mass, and imaging studies demonstrated an osteolytic lesion with cortical bone destruction and marked fluorodeoxyglucose uptake on positron emission tomography-computed tomography, raising a strong suspicion of malignancy. Histopathological examination revealed foreign-body granulomatous inflammation with characteristic crystal deposition, and the diagnosis of PCDD was definitively established through the combined use of gold hydroxamic acid staining, Raman spectroscopy, and ultrastructural analysis. Although surgical excision with curettage was initially achieved, local recurrence was observed 6 years later, indicating the potential for long-term disease persistence. In addition, a comprehensive literature review conducted in accordance with the PRISMA guidelines was performed to summarize previously reported cases of PCDD, with particular attention to anatomical distribution, radiological characteristics, recurrence patterns, and proposed pathogenic mechanisms. The review confirmed the extreme rarity of mandibular involvement and demonstrated that recurrence can occur apparently even after surgical treatment. Conclusions: This case underscores the importance of a multimodal diagnostic strategy integrating imaging, histopathology, and spectroscopic analyses for the accurate identification of PCDD and highlights the necessity of histopathological confirmation to prevent unnecessary aggressive treatment.<\/jats:p>","DOI":"10.3390\/diagnostics16040567","type":"journal-article","created":{"date-parts":[[2026,2,13]],"date-time":"2026-02-13T16:09:32Z","timestamp":1770998972000},"page":"567","update-policy":"https:\/\/doi.org\/10.3390\/mdpi_crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Recurrent Phosphoglyceride Crystal Deposition Disease in the Mandible Mimicked a Malignant Tumor: Insights from a Clinical Case Report and Literature Review"],"prefix":"10.3390","volume":"16","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-9345-4153","authenticated-orcid":false,"given":"Jumpei","family":"Shirakawa","sequence":"first","affiliation":[{"name":"Department of Oral and Maxillofacial Surgery, Kochi Medical School, Kochi University, 185-1, Kohasu, Oko-Cho, Nankoku-City 783-8505, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-5686-0243","authenticated-orcid":false,"given":"Motoo","family":"Ito","sequence":"additional","affiliation":[{"name":"Kochi Institute for Core Sample Research, X-Star, Japan Agency for Marine-Earth Science Technology (JAMSTEC), B200 Monobe, Nankoku-City 783-8502, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Takuya","family":"Matsuzaki","sequence":"additional","affiliation":[{"name":"Marine Core Research Institute, Kochi University, B200 Monobe, Nankoku-City 783-8502, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Mitsuko","family":"Iguchi","sequence":"additional","affiliation":[{"name":"Diagnostic Pathology Department, Kochi Medical School Hospital, 185-1, Kohasu, Oko-Cho, Nankoku-City 783-8505, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Kie","family":"Nakatani","sequence":"additional","affiliation":[{"name":"Department of Oral and Maxillofacial Surgery, Kochi Medical School, Kochi University, 185-1, Kohasu, Oko-Cho, Nankoku-City 783-8505, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Eri","family":"Sasabe","sequence":"additional","affiliation":[{"name":"Department of Oral and Maxillofacial Surgery, Kochi Medical School, Kochi University, 185-1, Kohasu, Oko-Cho, Nankoku-City 783-8505, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Yukio","family":"Yoshioka","sequence":"additional","affiliation":[{"name":"Department of Oral and Maxillofacial Surgery, Kochi Medical School, Kochi University, 185-1, Kohasu, Oko-Cho, Nankoku-City 783-8505, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Tetsuya","family":"Yamamoto","sequence":"additional","affiliation":[{"name":"YAMAKIN Next Generation Dental Medicine Development Course, Kochi Medical School, Kochi University, 185-1, Kohasu, Oko-Cho, Nankoku-City 783-8505, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Kenji","family":"Yamagata","sequence":"additional","affiliation":[{"name":"Department of Oral and Maxillofacial Surgery, Kochi Medical School, Kochi University, 185-1, Kohasu, Oko-Cho, Nankoku-City 783-8505, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"1968","published-online":{"date-parts":[[2026,2,13]]},"reference":[{"key":"ref_1","first-page":"508","article-title":"Unusual huge intramuscular granuloma with calcium phosphate crystal deposition in the buttock","volume":"42","author":"Kubo","year":"1992","journal-title":"Acta Pathol. Jpn."},{"key":"ref_2","doi-asserted-by":"crossref","first-page":"115296","DOI":"10.1016\/j.bcp.2022.115296","article-title":"Regulation of membrane phospholipid biosynthesis in mammalian cells","volume":"206","author":"Morita","year":"2022","journal-title":"Biochem. Pharmacol."},{"key":"ref_3","doi-asserted-by":"crossref","first-page":"807","DOI":"10.1016\/S0307-742X(21)00263-0","article-title":"Crystal deposition disease and the soft tissues","volume":"5","author":"Dieppe","year":"1979","journal-title":"Clin. Rheum. Dis."},{"key":"ref_4","doi-asserted-by":"crossref","first-page":"992","DOI":"10.1046\/j.1440-1827.2000.01138.x","article-title":"Phosphoglyceride crystal deposition disease","volume":"50","author":"Miura","year":"2000","journal-title":"Pathol. Int."},{"key":"ref_5","doi-asserted-by":"crossref","first-page":"881","DOI":"10.1111\/j.1440-1827.2004.01776.x","article-title":"Phosphoglyceride (PG) crystal deposition disease: A novel acquired disease in which PG crystals are deposited in injured soft tissue and bone with phospholipid metabolism disturbance","volume":"54","author":"Miura","year":"2004","journal-title":"Pathol. Int."},{"key":"ref_6","doi-asserted-by":"crossref","first-page":"8304","DOI":"10.12998\/wjcc.v10.i23.8304","article-title":"Phosphoglyceride crystal deposition disease requiring differential diagnosis from malignant tumors and confirmed by Raman spectroscopy: A case report","volume":"10","author":"Ohkura","year":"2022","journal-title":"World J. Clin. Cases"},{"key":"ref_7","doi-asserted-by":"crossref","first-page":"55","DOI":"10.1111\/pin.13290","article-title":"Phosphoglyceride crystal deposition disease in the intestine","volume":"73","author":"Tsutsumi","year":"2023","journal-title":"Pathol. Int."},{"key":"ref_8","doi-asserted-by":"crossref","first-page":"1413","DOI":"10.1023\/A:1015351320907","article-title":"Phosphoglyceride crystal deposition disease presenting as multiple paragastric masses","volume":"47","author":"Yachida","year":"2002","journal-title":"Dig. Dis. Sci."},{"key":"ref_9","doi-asserted-by":"crossref","first-page":"508","DOI":"10.1016\/j.jtcvs.2007.04.007","article-title":"Phosphoglyceride crystal deposition disease originating from the myocardium","volume":"134","author":"Shoji","year":"2007","journal-title":"J. Thorac. Cardiovasc. Surg."},{"key":"ref_10","doi-asserted-by":"crossref","first-page":"54","DOI":"10.1097\/RLU.0000000000005543","article-title":"High 18F-FDG uptake in phosphoglyceride crystal deposition disease of the humerus mimicking chondrosarcoma","volume":"50","author":"Matsutani","year":"2025","journal-title":"Clin. Nucl. Med."},{"key":"ref_11","doi-asserted-by":"crossref","first-page":"790","DOI":"10.1016\/j.recesp.2021.06.016","article-title":"The PRISMA 2020 statement: An updated guideline for reporting systematic reviews","volume":"74","author":"Page","year":"2021","journal-title":"Rev. Esp. Cardiol."},{"key":"ref_12","doi-asserted-by":"crossref","first-page":"347","DOI":"10.1111\/j.1471-4159.1959.tb12641.x","article-title":"The histochemical identification of myelin phosphoglycerides by their ferric hydroxamates","volume":"3","author":"Adams","year":"1959","journal-title":"J. Neurochem."},{"key":"ref_13","doi-asserted-by":"crossref","first-page":"125","DOI":"10.1111\/j.1471-4159.1963.tb11472.x","article-title":"The histochemical identification of phosphoglycerides in myelin","volume":"10","author":"Gallyas","year":"1963","journal-title":"J. Neurochem."},{"key":"ref_14","doi-asserted-by":"crossref","unstructured":"Hori, Y., Iwamoto, T., Ohkura, Y., Tsutsumi, N., Miura, K., Yachida, S., and Nakahara, O. (2023). Phosphoglyceride crystal deposition disease of the scapula: A case report and literature review. JBJS Case Connect., 13.","DOI":"10.2106\/JBJS.CC.22.00520"},{"key":"ref_15","doi-asserted-by":"crossref","first-page":"45","DOI":"10.1186\/s13000-025-01641-2","article-title":"A case of phosphoglyceride crystal deposition disease in the maxilla","volume":"20","author":"Saitou","year":"2025","journal-title":"Diagn. Pathol."},{"key":"ref_16","doi-asserted-by":"crossref","first-page":"2596","DOI":"10.1093\/eurheartj\/ehaa289","article-title":"Phosphoglyceride crystal deposition disease as a rare tumour after cardiac surgery","volume":"41","author":"Sato","year":"2020","journal-title":"Eur. Heart J."},{"key":"ref_17","doi-asserted-by":"crossref","first-page":"e315","DOI":"10.1016\/j.athoracsur.2020.07.099","article-title":"Complete resection of phosphoglyceride crystal deposition disease after cardiac surgery","volume":"111","author":"Miki","year":"2021","journal-title":"Ann. Thorac. Surg."},{"key":"ref_18","doi-asserted-by":"crossref","first-page":"16","DOI":"10.1016\/j.ejro.2018.01.004","article-title":"Phosphoglyceride crystal deposition disease mimicking a malignant tumor","volume":"5","author":"Tokue","year":"2018","journal-title":"Eur. J. Radiol. Open"},{"key":"ref_19","doi-asserted-by":"crossref","first-page":"860","DOI":"10.1111\/pin.13169","article-title":"Phosphoglyceride crystal deposition disease in a rib bone and ovary mimicking malignancy: A report of two cases including a previously undescribed cystic case","volume":"71","author":"Ishida","year":"2021","journal-title":"Pathol. Int."},{"key":"ref_20","doi-asserted-by":"crossref","first-page":"230","DOI":"10.1016\/j.jaccas.2021.09.024","article-title":"Severe functional tricuspid stenosis due to phosphoglyceride crystal deposition disease in right atrium","volume":"4","author":"Takeuchi","year":"2022","journal-title":"JACC Case Rep."},{"key":"ref_21","doi-asserted-by":"crossref","first-page":"830","DOI":"10.1016\/j.pathol.2021.10.007","article-title":"Phosphoglyceride crystal deposition disease: A rare pseudosarcomatous growth with peculiar predilections","volume":"54","author":"Omar","year":"2022","journal-title":"Pathology"},{"key":"ref_22","doi-asserted-by":"crossref","first-page":"112","DOI":"10.1097\/01.bsd.0000127701.37593.03","article-title":"Phosphoglyceride crystal deposition disease in the spine: A case report","volume":"18","author":"Nishimura","year":"2005","journal-title":"J. Spinal Disord. Tech."},{"key":"ref_23","doi-asserted-by":"crossref","first-page":"120","DOI":"10.1186\/s40792-018-0516-2","article-title":"Phosphoglyceride crystal deposition disease in the abdominal wall: A case report","volume":"4","author":"Nakahara","year":"2018","journal-title":"Surg. Case Rep."},{"key":"ref_24","doi-asserted-by":"crossref","first-page":"440","DOI":"10.1007\/s13691-024-00706-1","article-title":"Phosphoglyceride crystal deposition with suspected malignant ovarian tumor: A case report and literature review","volume":"13","author":"Furuzono","year":"2024","journal-title":"Int. Cancer Conf. J."},{"key":"ref_25","first-page":"751582","article-title":"A case of phosphoglyceride crystal deposition disease in the pelvic soft tissues recurring after initial surgery","volume":"2015","author":"Yamada","year":"2015","journal-title":"Case Rep. Obstet. Gynecol."},{"key":"ref_26","doi-asserted-by":"crossref","first-page":"442","DOI":"10.32074\/1591-951X-193","article-title":"Phosphoglyceride crystal deposition disease involving adnexa uteri: A case report with histogenetic consideration","volume":"113","author":"Nakamura","year":"2021","journal-title":"Pathologica"},{"key":"ref_27","doi-asserted-by":"crossref","first-page":"2026","DOI":"10.2967\/jnumed.117.197988","article-title":"Appropriate use criteria for 18F-FDG PET\/CT in restaging and treatment response assessment of malignant disease","volume":"58","author":"Jadvar","year":"2017","journal-title":"J. Nucl. Med."},{"key":"ref_28","first-page":"256","article-title":"Usefulness of MR imaging for odontogenic tumors","volume":"131","author":"Kurabayashi","year":"2021","journal-title":"Oral. Surg. Oral. Med. Oral. Pathol. Oral. Radiol."},{"key":"ref_29","doi-asserted-by":"crossref","first-page":"3134","DOI":"10.1002\/jbm.b.34639","article-title":"CD68+ macrophages as crucial components of the foreign body reaction demonstrate an unconventional pattern of functional markers quantified by analysis with double fluorescence staining","volume":"108","author":"Klinge","year":"2020","journal-title":"J. Biomed. Mater. Res. B Appl. Biomater."},{"key":"ref_30","doi-asserted-by":"crossref","first-page":"3284","DOI":"10.1021\/acs.chemrev.3c00608","article-title":"The chemical reactivity of membrane lipids","volume":"124","author":"Sanderson","year":"2024","journal-title":"Chem. Rev."},{"key":"ref_31","doi-asserted-by":"crossref","first-page":"413","DOI":"10.3109\/01913129209057826","article-title":"Ultrastructural analysis of asteroid bodies: Evidence for membrane lipid bilayer nature of components","volume":"16","author":"Papadimitriou","year":"1992","journal-title":"Ultrastruct. Pathol."},{"key":"ref_32","doi-asserted-by":"crossref","first-page":"15","DOI":"10.3109\/09687689509038490","article-title":"Micro-, nano- and meso-scale heterogeneity of lipid bilayers and its influence on macroscopic membrane properties","volume":"12","author":"Mouritsen","year":"1995","journal-title":"Mol. Membr. Biol."},{"key":"ref_33","doi-asserted-by":"crossref","first-page":"243","DOI":"10.1146\/annurev.bi.43.070174.001331","article-title":"Phosphoglyceride metabolism","volume":"43","year":"1974","journal-title":"Annu. Rev. Biochem."},{"key":"ref_34","doi-asserted-by":"crossref","first-page":"175","DOI":"10.1146\/annurev.pharmtox.39.1.175","article-title":"Regulation and inhibition of phospholipase A2","volume":"39","author":"Balsinde","year":"1999","journal-title":"Annu. Rev. Pharmacol. Toxicol."},{"key":"ref_35","doi-asserted-by":"crossref","first-page":"383","DOI":"10.1016\/j.rdc.2006.02.001","article-title":"Calcium crystal deposition diseases: Update on pathogenesis and manifestations","volume":"32","author":"Molloy","year":"2006","journal-title":"Rheum. Dis. Clin. N. Am."},{"key":"ref_36","doi-asserted-by":"crossref","first-page":"31","DOI":"10.1007\/s12325-014-0175-z","article-title":"A review of uric acid, crystal deposition disease, and gout","volume":"32","author":"Dalbeth","year":"2015","journal-title":"Adv. Ther."},{"key":"ref_37","doi-asserted-by":"crossref","unstructured":"Hegazi, A. (2023). Hydroxyapatite deposition disease: A comprehensive review of pathogenesis, radiological findings, and treatment strategies. Diagnostics, 13.","DOI":"10.3390\/diagnostics13162678"}],"container-title":["Diagnostics"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/www.mdpi.com\/2075-4418\/16\/4\/567\/pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,2,24]],"date-time":"2026-02-24T09:27:00Z","timestamp":1771925220000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.mdpi.com\/2075-4418\/16\/4\/567"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,2,13]]},"references-count":37,"journal-issue":{"issue":"4","published-online":{"date-parts":[[2026,2]]}},"alternative-id":["diagnostics16040567"],"URL":"https:\/\/doi.org\/10.3390\/diagnostics16040567","relation":{},"ISSN":["2075-4418"],"issn-type":[{"value":"2075-4418","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,2,13]]}}}