Phosphoglyceride crystal deposition disease involving adnexa uteri: a case report with histogenetic consideration

We present a rare case of phosphoglyceride crystal deposition disease (PCDD), as a gynecologic disease, with reference to histogenesis of crystal deposition. An 84-year-old woman, who had undergone simple hysterectomy for uterine leiomyoma 44 years previously, presented with multiple masses in the bilateral adnexa and the pelvic wall. The bilateral adnexal tumors were resected. The masses histologically revealed a foreign-body granuloma composed of numerous tiny, radially arranged needle-like crystal lumps surrounded by multinucleated giant cells and macrophages. The crystals showed birefringence under polarized light and were positive for gold hydroxamic acid stain, and the tumor was thus diagnosed as PCDD. The masses revealed central cystic changes due to old hemorrhage, which contained crystal lumps without foreign-body reaction or birefringence. The present case demonstrated for the first time that phosphoglyceride crystals developed in old hemorrhagic foci, although it was not confirmed whether the old hemorrhagic foci were formed after hysterectomy or due to endometriosis.