Journal
Pulmonary Tumor Thrombotic Microangiopathy Caused by Metastatic Ovarian Cancer: An Antemortem Diagnosis with Pulmonary Aspiration Cytopathology
A 48-year-old woman with advanced ovarian cancer was diagnosed with pulmonary tumor thrombotic microangiopathy (PTTM) by antemortem pulmonary wedge aspiration cytopathology. Despite the initiation of anti-cancer treatment, she unfortunately died due to progressive respiratory failure. Histopathology of the autopsied lung revealed multiple tumor embolization with fibrin-rich clot and fibro-cellular intimal proliferation at the pulmonary arteriole. The embolized tumor showed strong immune-positivity for pro-thrombotic and fibrotic factors (tissue factor and vascular endothelial growth factor), suggesting the underlying mechanisms of PTTM development. This case suggests that a quick antemortem diagnosis and the early induction of specific treatments might ensure a better prognosis of PTTM.
Pulmonary Artery Sarcoma Diagnosed Using an Endovascular Catheter Forceps Biopsy
We herein report a case of pulmonary artery sarcoma (PAS) in a 64-year-old woman. She was admitted to our hospital because of massive genital bleeding from endometrial cancer. Contrast-enhanced computed tomography (CT) revealed a left pulmonary artery mass and deep vein thrombosis. She underwent anticoagulant therapy for one year. However, the mass lesion gradually expanded.
Two Cases of Encephalitis without Anti-N-methyl-D-aspartate Receptor Antibody Successfully Treated with Ovarian Teratoma Resection and Immunotherapy
Anti-N-methyl-D-aspartate receptor (NMDAR) encephalitis is an autoimmune disorder sometimes accompanied by ovarian teratoma. However, the concept of encephalitis without anti-NMDAR antibodies successfully treated with ovarian teratoma resection and immunotherapy has not been established. We herein report two such cases. Case 1 exhibited delayed magnetic resonance imaging abnormalities in the thalamus and basal ganglia, despite clinical improvement. Case 2 presented with brainstem encephalitis similar to Bickerstaff's encephalitis. Although both patients tested negative for anti-NMDAR antibodies, the recovery of the neurological function and good prognosis following tumor resection with immunotherapy indicated a close association between these diseases and ovarian teratoma.
Remitting Seronegative Symmetrical Synovitis with Pitting Edema Syndrome as a Manifestation of Recurrent Ovarian Cancer
We herein report a case of ovarian cancer recurrence detected every time with symptoms of remitting seronegative symmetrical synovitis with pitting edema (RS3PE) syndrome. A 46-year-old woman who had a history of ovarian cancer 9 months earlier developed joint pain along with pitting edema in both hands and was diagnosed with RS3PE syndrome. Two and four years after initial surgery for ovarian cancer, symptoms of RS3PE syndrome appeared, and a recurrent site was detected. With resection of the relapsed sites and increased maintenance dose of methylprednisolone, these symptoms improved within a month.
Ovarian Follicular Lymphoma Diagnosed Due to Hydronephrosis
A 74-year-old woman presented with left lateral abdominal pain. Abdominal echography revealed left hydronephrosis and a pelvic mass. The patient underwent left adnexal resection of a suspected left ovarian tumor and was diagnosed with follicular lymphoma (FL) of clinical stage IIIA, grade 2. The patient was treated with rituximab-combined chemotherapy and achieved complete remission. The most common histological types of ovarian lymphoma are diffuse large B-cell lymphoma and Burkitt lymphoma, with FL being an extremely rare variant. We herein report a case of ovarian FL diagnosed as hydronephrosis.
Ovarian Microcystic Stromal Tumor with Significant Nestin Expression: A Unique Case
Ovary microcystic stromal tumor (MCST) is an extremely rare subtype of sex cord-stromal neoplasm, and only 57 cases have been reported. We herein report a unique case of ovarian MCST with positive nestin expression in a 39-year-old Chinese woman. The tumor showed microcystic stromal histological structures and characteristically expressed the CD10, WT-1, and Ki67 proteins. A molecular analysis identified a point mutation (c.110C > T) in exon 3 of the CTNNB1 gene. To our knowledge, no report has described a case of ovarian MCST with positive staining for nestin protein. Our study provides new insights into the tumor biology of ovarian MCST.
Immune Checkpoint Inhibitor-induced Mass-forming Pancreatitis and Lipodystrophy: A Case Report and Review of the Literature
Mass-forming pancreatitis and lipodystrophy are rare immune-related adverse events (irAEs). A 71-year-old woman received 12 cycles of pembrolizumab for recurrent uterine endometrial cancer. The patient presented with mass-forming pancreatitis and lipodystrophy. A needle biopsy of the pancreatic tissue revealed lymphocytic infiltration and fibrosis. Simultaneously, the patient exhibited lipodystrophy. The recurrent tumors disappeared, so the patient discontinued immune checkpoint inhibitor treatment and was administered oral prednisolone. In addition, the pancreatic mass disappeared; however, the fat loss did not improve. Considering pancreatitis and lipodystrophy to be irAEs, prompt management is important.
Ectopic Production of Parathyroid Hormone and Production of Parathyroid Hormone-related Protein in Dedifferentiated Endometrial Carcinoma Induced Severe Hypercalcemia
Hypercalcemia is a significant complication in cancer patients, primarily caused by parathyroid hormone-related peptide (PTHrP) and, rarely, by parathyroid hormone (PTH) production from tumors. We report a case of severe hypercalcemia in a woman with uterine cancer who exhibited elevated PTH and PTHrP levels. Surgical intervention revealed dedifferentiated endometrial carcinoma. Postoperatively, PTH and PTHrP levels normalized but subsequently increased due to metastases. A molecular analysis confirmed the expression of the PTH gene and protein within the tumor, indicating ectopic PTH production. In diagnosing and treating cancers, it is necessary to consider not only PTHrP production but also ectopic PTH production.
Anti-<i>N</i>-methyl-d-aspartate Receptor Encephalitis Caused by a Teratoma Arising From the Retroperitoneum
Radiation Nephropathy Complicated by Tubulointerstitial Nephritis with Predominantly Lymphocyte and Plasma Cell Infiltration
A 64-year-old Japanese woman presented with gross hematuria and was diagnosed with stage IVB cervical cancer. Renal dysfunction was observed following chemoradiation therapy, and a renal biopsy revealed renal thrombotic microangiopathy (TMA) and tubulointerstitial nephritis (TIN), which are atypical features of radiation nephropathy. Contrast-enhanced computed tomography revealed wedge-shaped areas of high contrast, consistent with areas of high radiation dose in the kidneys, thus leading to the diagnosis of radiation nephropathy. This case underscores the importance of integrating clinical, pathological, and radiological findings for the diagnosis of radiation nephropathy, atypical renal biopsy findings, and a combination of TIN and renal TMA.
Peritoneal and Ovarian Tuberculosis with Massive Ascites
Extremely Late Recurrence of Ovarian Carcinoma Diagnosed by an Endoscopic Ultrasound-guided Fine-needle Biopsy
We herein report a case of recurrence of epithelial ovarian carcinoma 41 years after the primary surgery that was diagnosed by an endoscopic ultrasound-guided fine-needle biopsy (EUS-FNB). The differential diagnosis based on the imaging findings was difficult. We performed an EUS-FNB and compared the EUS-FNB specimen to the surgical specimen that had been resected in the primary surgery for ovarian carcinoma 41 years earlier, including immunohistochemical staining. Finally, we made a definitive diagnosis of extremely late recurrence of ovarian carcinoma of the retroperitoneum. An EUS-FNB enables an accurate histological diagnosis by obtaining a sample that is large enough to perform immunohistochemical staining.
Remote Recurrence from Ovarian Cancer Mimicking Lung Cancer
Pneumocystis Pneumonia in a Patient with Ovarian Cancer Receiving Olaparib Therapy
We herein report a case of pneumocystis pneumonia (PCP) in a 77-year-old woman with ovarian cancer who was receiving olaparib therapy. After the patient's second relapse of ovarian cancer, she was administered olaparib as maintenance therapy following successful completion of docetaxel and carboplatin therapy. On receiving olaparib, she showed symptoms of a fever and malaise. Based on laboratory and imaging findings, she was diagnosed with PCP. After treatment with corticosteroids and trimethoprim/sulfamethoxazole followed by atovaquone, the patient's general condition improved. The lymphocytopenia observed after olaparib administration may have been associated with the development of PCP.
Cervical Skeletal Muscle Metastasis of Colorectal Cancer
Anti-nuclear Matrix Protein 2 Antibody-positive Dermatomyositis Associated with Cervical Cancer Recurrence after 6 Years of Stable Disease
A 79-year-old woman was evaluated for weakness, dysphagia, and elevated levels of creatinine kinase. Her medical history included stage IIIB cervical cancer eight years previously, which improved after undergoing radiotherapy. Two years later, cancer recurred in the right hilar and mediastinal lymph nodes, and the patient was successfully treated with chemotherapy. Physical examinations showed Gottron's sign, the V sign, the Holster sign, and nailfold erythema. Computed tomography revealed left supraclavicular lymphadenopathy, compatible with cancer recurrence. Anti-nuclear matrix protein 2 (NXP2) antibody was found in her sera and a diagnosis of cancer-associated dermatomyositis was thus made. This case suggests that cancer progression over a period of years may have triggered the onset of autoimmunity against NXP2.
Atypical Image Change of Hepatic Mucinous Cystic Neoplasm
KL-6 as a Tumor Marker of Primary Peritoneal Cancer in Patients with Connective Tissue Diseases
Paraneoplastic Inflammatory Myositis False-positive for Anti-aminoacyl-tRNA synthetase Antibody Successfully Ameliorated by Immunosuppressive Therapy
We herein report a 60-year-old woman with anti-aminoacyl-tRNA synthetase (ARS) antibody-false-positive paraneoplastic polymyositis complicated with ovarian carcinoma, which was promptly ameliorated by immunosuppressive therapy, including glucocorticoids (GCs), before treatment against malignancy. The patient was admitted to our hospital because of rapidly progressive muscular weakness of the neck and both limbs over three weeks. Upon a detailed examination, the patient tested positive for anti-ARS antibodies by an enzyme-linked immunosorbent assay (ELISA), which were later determined to be false positives, as confirmed by immunoblotting and immunoprecipitation. She was strongly suspected to have ovarian carcinoma and pulmonary metastasis. A biopsy of her muscle revealed myositis, mainly with CD68
Acute Myocardial and Hemorrhagic Cerebral Infarction Secondary to Non-bacterial Thrombotic Endocarditis in a Patient with Advanced Uterine and Ovarian Cancer
Non-bacterial thrombotic endocarditis (NBTE) causes myocardial and cerebral infarctions and is associated with advanced stages of malignancy. However, only a few cases of myocardial and cerebral infarctions have been reported in the same patient. We herein report a 47-year-old woman with advanced uterine and ovarian cancer who experienced acute myocardial infarction (MI) after receiving chemotherapeutic intervention for the cancer and hemorrhagic cerebral infarction 1 month after admission for acute MI, attributable to NBTE of the aortic valve. NBTE should be considered in patients with malignancies and life-threatening thromboembolism of important organs.
Ovarian Cancer with TTF-1-positive Giant Pleural Dissemination
Thyroid transcription factor 1 (TTF-1) is primarily expressed in lung and thyroid cancers, but it has also been observed in other cancers. A 60-year-old woman presented with worsening dyspnea, pleural effusion, lung metastases, a right ovarian tumor, and para-aortic intra-abdominal lymph node metastasis. Biopsies from the pleural dissemination revealed TTF-1-positive adenocarcinomas. To confirm the presence of the primary organ, biopsies were performed on a para-aortic intra-abdominal lymph node that ascended from the ovaries. An adenocarcinoma positive for TTF-1 was identified, thus confirming the diagnosis of ovarian cancer. Our findings revealed that TTF-1 positivity does not always signify a lung cancer origin.
Atypical Pseudo-Meigs' Syndrome without Pleural Effusion Due to Ovarian Metastasis from Sigmoid Colon Cancer
We herein report a case of atypical pseudo-Meigs' syndrome without pleural effusion. A 46-year-old woman was diagnosed with an ovarian tumor and sigmoid colon cancer with massive ascites. She underwent surgical resection of the sigmoid colon and bilateral salpingo-oophorectomy. The pathological diagnosis was sigmoid colon cancer with ovarian metastasis. A few days after the operation, the massive ascites disappeared. Immunostaining for vascular endothelial growth factor (VEGF) suggested its overproduction was involved in the development of the ascites. Although cases of pseudo-Meigs' syndrome without pleural effusion are rare, reporting such cases will facilitate the choice of more appropriate treatment strategies in future.
Japanese Society of Internal Medicine
0918-2918