Anti-transcription intermediary factor 1 gamma (TIF1γ) antibody-positive dermatomyositis associated with ascending colon cancer: a case report and review of the literature.

BACKGROUND: Anti-transcriptional intermediary factor 1 gamma (TIF1γ) antibody is a marker for predicting cancer association in patients with dermatomyositis (DM). The overall survival rate in DM patients with cancer was reported to be considerably worse than that in DM patients without cancer. However, the treatment for cancer-associated DM remains controversial, because the treatment priority between surgical resection for the tumor and internal treatments, including glucocorticoids, immunosuppressive agents, and intravenous immune globulin, has not been established. CASE PRESENTATION: We report the case of a 57-year-old Japanese man diagnosed with anti-TIF1γ antibody-positive DM associated with ascending colon cancer. His clinical symptoms included facial and brachial edema, muscle weakness, dysphagia, myalgia, and rash. Physical examination revealed periorbital edema and Gottron's papules over his knuckles with brachial edema, and tenderness and weakness of the proximal limb muscles. The findings of hyperintense muscles in T2-weighted sequences of brachial contrast-enhanced magnetic resonance imaging and the infiltration of lymphocytic cells and CD4-positive lymphocytes from muscle biopsy were compatible with the diagnostic criteria for dermatomyositis. Anti-TIF1γ antibody was positive by immunoprecipitation assay. He first started internal treatment including intravenous immunoglobulin, steroid pulse, prednisolone, and azathioprine, followed by surgical resection for the tumor because of the elevation of creatine kinase and progression of dysphagia. However, clinical symptoms did not improve, and the patient died 6 months later. CONCLUSIONS: We faced difficulties in determining the treatment priority between surgical resection and internal treatment for our case; therefore, this case would be educational for readers. We searched PubMed to identify English-language case reports of anti-TIF1γ antibody-positive dermatomyositis with malignancy and found 21 reported cases. We herein review and summarize previously reported cases of anti-TIF1γ antibody-positive DM with malignancy. Cancer screening is essential in patients with anti-TIF1γ antibody-positive dermatomyositis because it is associated with a high prevalence of malignancies. Our review revealed that initial surgical treatment should be recommended for better prognosis if the general condition allows.

Angioimmunoblastic T-cell lymphoma accompanied by pure red cell aplasia: A case report.

BACKGROUND: Angioimmunoblastic T-cell lymphoma (AITL) is a peripheral T-cell lymphoma, which is a rare subtype of lymphoma. Patients with AITL often have skin lesions, which are observed in 50% of all cases; the chief complaint of this patient was palpable purpura. AITL often complicates autoimmune or hematological disorders; however, among these, pure red cell aplasia (PRCA) is a very rare complication of AITL. We herein report a case of AITL with PRCA. CASE SUMMARY: A 77-year-old Japanese man presented to our hospital with complaints of loss of appetite for 2 mo and a 10-d history of palpable purpura. On physical examination, the patient was afebrile but had bilateral multiple palpable purpuric lesions over the lower extremities, lower abdomen, and part of the upper extremities. Moreover, lymphadenopathy of the bilateral inguinal, cervical, and supraclavicular nodes was noted. Laboratory and imaging studies and skin biopsy were conducted but were inconclusive. Based on inguinal lymph node excisional biopsy, we diagnosed the patient with AITL. Subsequently, the patient developed progressive normocytic normochromic anemia that necessitated almost daily blood transfusion. The clinical presentations and results of bone marrow assessment were consistent with those of PRCA, which is associated with AITL. Chemotherapy was initiated but was not effective. The patient refused further chemotherapy and opted to continue receiving best supportive care. CONCLUSION: PRCA is an extremely rare complication of AITL. As the pathophysiology remains unclear, further research is warranted.

Hepatic methotrexate-associated lymphoproliferative disorders identified by multiple liver tumors: a case report and review of the literature.

BACKGROUND: Methotrexate, an immunosuppressant, is widely used as the standard therapeutic drug for rheumatoid arthritis. With the increasing frequency of use of methotrexate, adverse effects of methotrexate have been reported, one of which is known as methotrexate-associated lymphoproliferative disorders. The etiology of hepatic methotrexate-associated lymphoproliferative disorders remains largely unknown. To date, there have only been ten cases of hepatic methotrexate-associated lymphoproliferative disorders reported in the English literature and a case report is very rare. CASE PRESENTATION: An 82-year-old Japanese man with rheumatoid arthritis treated with methotrexate presented with fever. Contrast-enhanced computed tomography showed multiple hypovascular nodules in his liver, spleen, and lung, and para-aortic lesions. Endoscopic ultrasound-guided fine-needle aspiration biopsy for liver tumors was performed, and pathological results identified cluster of differentiation 20-positive lymphocytes. Discontinuance of methotrexate led to regression of the nodules and a final definitive diagnosis of methotrexate-associated lymphoproliferative disorders was made. CONCLUSIONS: We review 11 reported cases of hepatic methotrexate-associated lymphoproliferative disorders including the present case. Physicians should discontinue methotrexate in patients with rheumatoid arthritis treated with methotrexate when elevated soluble interleukin-2 receptor and hypovascular lesions in contrast-enhanced computed tomography are confirmed considering the possibility of methotrexate-associated lymphoproliferative disorders.

Hypothalamic hypopituitarism secondary to suprasellar metastases from small cell lung cancer: a case report and review of the literature.

BACKGROUND: Metastasis to the pituitary gland is an infrequent clinical problem, and the symptoms caused by metastases have been reported in only 2.5-18.2% of the cases. However, metastasis to the suprasellar lesion has rarely been reported in the literature. To the best of our knowledge, only nine cases of hypothalamic hypopituitarism due to metastases of solid tumors have been reported in English-language journals. CASE PRESENTATION: A 67-year-old Japanese man presented to our hospital with generalized weakness, lethargy, and weight loss. Laboratory data showed hypoglycemia together with low thyroid-stimulating hormone and free thyroxine. We suspected hypopituitarism and performed imaging of the head, which revealed multiple tumors, one of which was in the suprasellar region. Computed tomography of the chest showed a tumor shadow, and a bronchoscopic biopsy pathologically showed small cell lung cancer. Hormone profiling demonstrated hypothalamic pan-hypopituitarism. We diagnosed hypothalamic hypopituitarism secondary to metastases from the primary lung cancer and initiated radiation, chemotherapy, and hormone replacement, but the patient died 10 months later. CONCLUSIONS: We report a case of a 67-year-old man with hypothalamic hypopituitarism secondary to a suprasellar metastasis from a primary small cell lung cancer, and we review ten cases of hypothalamic hypopituitarism due to metastases, including our patient. Recognizing hypopituitarism can be challenging, especially in the elderly, whose symptoms such as lethargy and visual decline may be mistaken for the natural aging process. In patients with established metastatic conditions, the symptoms may be wrongly attributed to malignancy or to the side effects of therapy. When a patient is suspected of having hypopituitarism, a hormone load test can help to diagnose the type of hypopituitarism. It is important to evaluate the brain and the whole body to confirm whether metastasis and primary cancer exist. Because the mortality rate is very high, aggressive intervention for both diagnosis and therapy is required in cases of hypothalamic hypopituitarism secondary to tumor metastasis.

CT-guided Biopsy for the Diagnosis of Pulmonary Epithelioid Hemangioendothelioma Mimicking Metastatic Lung Cancer.

A 69-year-old male patient presented with multiple lung nodules revealed by chest-computed tomography (CT) during a preoperative examination for an appendiceal tumor. The nodule diameters ranged from 2-10 mm without either pleural thickening or effusions. A fluorine-18-labeled fluorodeoxyglucose (18F-FDG)-positron emission tomography (PET)/CT scan showed a high FDG uptake in the appendiceal tumor, but almost normal standardized uptake values in the bilateral lung nodules. A CT-guided biopsy led to a diagnosis of pulmonary epithelioid hemangioendothelioma, a rare vascular tumor with a radiological presentation similar to that of a metastatic lung tumor. The present case is the first to describe successful treatment using a CT-guided biopsy instead of more conventional methods.

Platypnea-orthodeoxia Syndrome Induced by an Infected Giant Hepatic Cyst.

An 83-year-old man was admitted with a chief complaint of exacerbation of dyspnea. His blood oxygen saturation was 90% in the recumbent position despite oxygen therapy, and it dropped to less than 80% when the patient attempted to sit upright. A computed tomography scan revealed a giant hepatic cyst compressing the right atrium and the inferior vena cava. After percutaneous drainage, the oxygen saturation improved and did not change with alteration of the patient's positions from recumbent to sitting or standing. This case report describes a patient with the platypnea-orthodeoxia syndrome due to a giant hepatic cyst successfully managed by percutaneous drainage.

Myositis Ossificans of the Hip Due to Pyogenic Arthritis Caused by Campylobacter fetus Subspecies fetus.

A 61-year-old woman was admitted with severe hip pain causing immobility and high serum levels of inflammatory markers. The patient had a medical history of diabetes. She had been scheduled to undergo right hip replacement surgery for the treatment of osteoarthritis associated with gradually progressive pain. On admission, an enhanced abdominal computed tomography scan showed an abnormal increase in synovial fluid surrounding the right ilium, with piriformis muscle calcification. Subsequent blood and specimen cultures identified Campylobacter fetus subspecies fetus. This is a rare report of a case of myositis ossificans followed by C. fetus pyogenic arthritis of the hip.