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Yoshinori Ohno, Teru Kumagi, Taira Kuroda, Mitsuhito Koizumi, Nobuaki ...
2014 Volume 53 Issue 11 Pages
1125-1129
Published: 2014
Released on J-STAGE: June 01, 2014
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Biliary drainage was performed in a 71-year-old man with obstructive jaundice of unknown origin; however, he died due to acute pulmonary failure. At autopsy, proliferation of adenocarcinoma cells was observed in the gallbladder mucosa transitioning from isolated signet-ring cell carcinoma (SRCC) to the subserosa and bile ducts without growth toward the gallbladder lumen. Furthermore, fibrocellular intimal proliferation, tumor emboli and organized thrombi were observed in the small pulmonary arteries. The final diagnosis was gallbladder carcinoma complicated by SRCC associated pulmonary tumor thrombotic microangiopathy (PTTM). PTTM may present as rapidly progressive dyspnea, and a high level of clinical suspicion is required to make the differential diagnosis.
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Hiroaki Kikuchi, Makoto Aoyagi, Kiyotaka Nagahama, Yu Yajima, Chisato ...
2014 Volume 53 Issue 11 Pages
1131-1135
Published: 2014
Released on J-STAGE: June 01, 2014
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A 76-year-old woman with a history of lumbar fracture and marked proteinuria, bilateral pitting edema, malaise and pruritus was referred for an evaluation of an impaired renal function. A renal biopsy led to a tentative diagnosis of acute interstitial nephritis (AIN) with minimal change disease caused by nonsteroidal anti-inflammatory drugs (NSAIDs). Following the discontinuation of oral NSAIDs, the patient's symptoms disappeared spontaneously. However, nephrotic-range proteinuria relapsed one month after discharge, following loxoprofen patch use. The withdrawal of the topical loxoprofen patches once again resulted in the disappearance of all symptoms. This is the first case report of nephrotic-range proteinuria and AIN secondary to topical NSAID patch use.
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Mina Gochi, Noboru Takayanagi, Takashi Ishiguro, Yosuke Miyahara, Tsut ...
2014 Volume 53 Issue 11 Pages
1137-1141
Published: 2014
Released on J-STAGE: June 01, 2014
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A 73-year-old woman with rheumatoid arthritis treated with methotrexate and prednisolone was admitted with dyspnea and ground-glass opacity on chest CT. We diagnosed her with
Pneumocystis jirovecii pneumonia (PCP) based on a positive PCR analysis of
Pneumocystis jirovecii and the presence of cysts in bronchoalveolar lavage fluid. The PaO
2 was 74.7 Torr on room air, and treatment with sulfamethoxazole-trimethoprim only was initiated. The hypoxemia and ground-glass opacity increased on hospital day 3, and the administration of adjunctive steroid therapy resulted in an improvement in the patient's condition. Although patients with PCP with HIV infection and hypoxemia are often treated with adjunctive steroid therapy to prevent adverse immune reactions, the efficacy of additive steroid administration in case of non-HIV PCP has not been established.
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Takashi Ishiguro, Noboru Takayanagi, Eriko Kawate, Yotaro Takaku, Naho ...
2014 Volume 53 Issue 11 Pages
1143-1147
Published: 2014
Released on J-STAGE: June 01, 2014
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A 76-year-old woman suffering from granulomatosis with polyangitis (GPA) developed organizing pneumonia with positive antineutrophil cytoplasmic antibodies and microscopic hematuria. Prednisolone improved the hematuria and radiological findings; however, after tapering the dose of prednisolone, a posterior left atrial wall mass was detected in association with a fever. Both regressed spontaneously, although secretory otitis media and sinusitis were noted; the resected sinusitis specimen exhibited vasculitis highly suggestive of GPA. The clinical picture of GPA with multi-organ involvement can vary. Recognizing the various manifestations of GPA is therefore necessary in order to provide an appropriate diagnosis and disease management.
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Hisao Imai, Kenji Inafuku, Kazushige Wakuda, Akira Ono, Tetsuhiko Tair ...
2014 Volume 53 Issue 11 Pages
1149-1152
Published: 2014
Released on J-STAGE: June 01, 2014
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We herein present the case of a 36-year-old woman who developed perianal metastasis of non-small cell lung cancer that was diagnosed based on the presence of symptoms mimicking a hemorrhoid. The patient initially underwent radiotherapy for a left superior sulcus tumor, then subsequently complained of a perianal mass that had prolapsed and bled. The tumor was removed via resection. Histologically, the mass was diagnosed as poorly differentiated carcinoma and considered to be a metastatic lesion arising from the primary lung cancer.
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Ahmet Emre Eskazan, Mehmet Sinan Dal, Safak Kaya, Tuba Dal, Orhan Ayyi ...
2014 Volume 53 Issue 11 Pages
1153-1158
Published: 2014
Released on J-STAGE: June 01, 2014
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Brucellosis is a worldwide zoonotic disease associated with hemolytic complications, including thrombotic microangiopathy (TMA) and hemolytic anemia. Autoimmune hemolytic anemia (AIHA) is a rare clinical presentation of this disease. In this report, we describe the cases of two patients with brucellosis who presented with Coombs-positive AIHA. We also include a review of the literature on the hemolytic complications of brucellosis. Both patients were successfully treated with a combination of doxycycline and rifampicin in addition to steroids. In the medical literature, there are several cases of TMA associated with brucellosis, although only a few cases of Coombs test-positive AIHA have been reported. Antibiotic therapy is the mainstay of treatment, and the selection of antibiotics and duration of treatment do not differ between brucellosis patients with and without hemolysis. Although rare, the potential for brucellosis should always be kept in mind in patients who present with hemolysis, especially those living in areas where brucellosis is endemic.
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Yusuke Isshiki, Chikako Ohwada, Emi Togasaki, Ryoh Shimizu, Nagisa Has ...
2014 Volume 53 Issue 11 Pages
1159-1164
Published: 2014
Released on J-STAGE: June 01, 2014
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Myeloid sarcoma (MS) is an extramedullary myeloid tumor that sometimes presents with antedating systemic leukemia, leading physicians to the misdiagnosis of lymphoma. CD25 is expressed in 13% of patients with acute myeloid leukemia (AML), and its expression is associated with
FLT3-ITD mutations, an elevated serum soluble interleukin 2 receptor (sIL-2R) level and a lower survival rate. However, there are no reports concerning the relationship between MS and the CD25 expression. We herein report a case of AML accompanied by thoracic epidural MS with a high CD25 expression, the
FLT3-ITD mutation and an extremely elevated serum sIL-2R level in a 59-year-old man who presented with paraplegia.
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Junya Kuroda, Yuji Shimura, Shinsuke Mizutani, Hisao Nagoshi, Miki Kiy ...
2014 Volume 53 Issue 11 Pages
1165-1169
Published: 2014
Released on J-STAGE: June 01, 2014
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Azacitidine is the first-line therapeutic option for myelodysplastic syndrome (MDS). This report describes a case of MDS in a patient who developed fatal acute interstitial pneumonitis (AIP) after the first seven-day course of intravenous azacitidine (75 mg/m
2/day) treatment. A review of previous and present studies of azacitidine-associated AIP suggests that azacitidine may cause life-threatening AIP during or after the first seven-day course of treatment, with pyrexia commonly preceding AIP. Although the non-hematologic adverse events associated with azacitidine are generally relatively mild, further accumulation of evidence may help to make an earlier diagnosis of azacitidine-associated AIP.
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Shinobu Tamura, Asumi Koyama, Chieko Shiotani, Toshio Kurihara, Akinor ...
2014 Volume 53 Issue 11 Pages
1171-1175
Published: 2014
Released on J-STAGE: June 01, 2014
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Primary plasma cell leukemia (PPCL) is a rare disease that progresses rapidly. In such cases, it is difficult to achieve remission, and early intensive chemotherapy is recommended. We herein describe the case of a 76-year-old man with PPCL complicated by renal failure and pulmonary hypertension. Bortezomib/dexamethasone induction therapy with lenalidomide was administered in association with continuous hemodiafiltration (CHDF). Complete remission was achieved after a single course of treatment, resulting in the cessation of CHDF. With the patient in remission, the administration of beraprost and bosentan resulted in improvements in the pulmonary hypertension. The results of this case report support the use of bortezomib/lenalidomide/dexamethasone combination therapy as an effective treatment for elderly PPCL patients with various complications.
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Yosuke Matsumoto, Shigeo Horiike, Saori Maekawa, Taro Isohisa, Natsumi ...
2014 Volume 53 Issue 11 Pages
1177-1181
Published: 2014
Released on J-STAGE: June 01, 2014
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This report concerns a 62-year-old man with primary cutaneous diffuse large B-cell lymphoma (PCLBCL), leg type that developed during methotrexate (MTX) treatment for rheumatoid arthritis (RA). Several tumors were observed on the left lower leg. A histological analysis showed diffuse proliferation of large neoplastic B-cells that were immunophenotypically CD10-/MUM1+/BCL6-/BCL2+ and cytogenetically had
IgH/c-MYC translocation without translocation involving
BCL6 or
IgH/BCL2. No evidence of Epstein-Barr virus (EBV) infection was found. The discontinuation of MTX resulted in a 20-month disease-free period. No previous cases of PCLBCL, leg type associated with RA or MTX therapy have been reported. The phenotypes of our patient were partly different from those of typical PCLBCL, leg type or RA/MTX-associated lymphoma.
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Makoto Hibino, Shigeto Horiuchi, Yoichi Okubo, Takuya Kakutani, Motoki ...
2014 Volume 53 Issue 11 Pages
1183-1185
Published: 2014
Released on J-STAGE: June 01, 2014
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We herein report the case of a previously healthy 24-year-old Japanese woman who developed adult-onset clinically mild encephalitis/encephalopathy with a reversible splenial lesion (MERS) presenting with hemiparesis and hemianesthesia secondary to adenovirus infection. The patient's neurological symptoms and the lesion in the splenium resolved within 17 days without therapy. The radiographic features and clinical course observed in this case were consistent with a diagnosis of MERS; however, the only neurological symptoms were hemiparesis and hemianesthesia. This is the first reported case of MERS involving only hemiparesis and hemianesthesia at onset. This case suggests that a diagnosis of MERS should be suspected in patients with hemiparesis and hemianesthesia, especially when these conditions are preceded by infection.
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Natsuko Yuki, Akira Yoshioka, Rei Yasuda, Takuma Ohmichi, Nobuyuki Oka
2014 Volume 53 Issue 11 Pages
1187-1190
Published: 2014
Released on J-STAGE: June 01, 2014
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We herein report the case of a patient with hepatitis C virus (HCV)-associated neuropathy with atypical pathological findings of a biopsied sural nerve. A 48-year-old man was admitted for a gait disturbance. Purpura and edema on the legs and hyperalgesia on the distal extremities were noted. The plasma HCV viral load was high, and cryoglobulin was positive. In the biopsied sural nerve, perivascular eosinophilic infiltration was associated with extravascular granuloma formation in the epineurium. The patient's symptoms disappeared following treatment with interferon-α and ribavirin. The present case suggests that HCV infection can lead to peripheral neuropathy associated with eosinophilic infiltration and granuloma formation.
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Atsushi Niwa, Yoko Okamoto, Takayuki Kondo, Hidehiko Nabatame, Ryosuke ...
2014 Volume 53 Issue 11 Pages
1191-1195
Published: 2014
Released on J-STAGE: June 01, 2014
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We herein report an autopsy case of relapsing polychondritis encephalitis coexisting with a Lewy body pathology and also review previous autopsy cases. A 59-year-old man exhibited a tremor of the right hand, small-steppage gait and bradykinesia. Five years later, he presented with relapsing auricular chondritis and scleritis and subsequently showed exacerbation of extrapyramidal symptoms. A histological examination revealed perivascular lymphocytic cuffing and infiltration in the small vessels, as well as loss of nerve cells and gliosis in the basal ganglia, insular gyrus and medial temporal lobe. The present case was characterized by perivasculitic panencephalitis and the coexistence of a Lewy body pathology, which may have augmented the patient's parkinsonism.
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Yuji Kato, Takeshi Hayashi, Akiko Kawai-Masaoka, Takaya Ichimura, Atsu ...
2014 Volume 53 Issue 11 Pages
1197-1200
Published: 2014
Released on J-STAGE: June 01, 2014
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A 40-year-old man visited to our hospital due to progressive right hemiparesis. Magnetic resonance imaging demonstrated a heterogeneous contrast-enhanced lesion in the left basal ganglia with compression of the ventricles. A brain biopsy did not demonstrate central nervous system (CNS) lymphoma, although acute demyelination was observed. Despite the administration of steroids, the lesion increased in size, and the patient died three months after admission. An autopsy disclosed perivascular and parenchymal infiltration of lymphoma cells. An immunohistochemical analysis showed that the lesion was a cytotoxic T-cell lymphoma. This case indicates that the development of primary CNS lymphoma of this immunophenotype may be preceded by demyelination with subsequent rapid progression, thus requiring a careful evaluation and meticulous diagnosis.
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Ichiro Nozaki, Yutaka Furukawa, Yuko Kato-Motozaki, Tokuhei Ikeda, Ats ...
2014 Volume 53 Issue 11 Pages
1201-1204
Published: 2014
Released on J-STAGE: June 01, 2014
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We herein describe the case of an 81-year-old Japanese woman with neuroleptic malignant syndrome that occurred 36 days after the initiation of combination therapy with tiapride (75 mg/day) and tetrabenazine (12.5 mg/day) for Huntington's disease. The patient had been treated with tiapride or tetrabenazine alone without any adverse effects before the administration of the combination therapy. She also had advanced breast cancer when the combination therapy was initiated. To the best of our knowledge, the occurrence of neuroleptic malignant syndrome due to combination therapy with tetrabenazine and tiapride has not been previously reported. Tetrabenazine should be administered very carefully in combination with other neuroleptic drugs, particularly in patients with a worsening general condition.
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Takeshi Yoshida, Yukio Tezuka, Takanobu Hirosawa, Hiroshi Umeki, Haruo ...
2014 Volume 53 Issue 11 Pages
1205-1208
Published: 2014
Released on J-STAGE: June 01, 2014
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A 62-year-old man was admitted to our hospital complaining of dysphagia and hoarseness that had persisted for five days. A neurological examination indicated bulbar palsy. Brain magnetic resonance imaging showed thickening of cranial nerves IX, X and XI, in addition to pineal body enlargement with diffuse contrast enhancement. A tumor biopsy overriding the spinal root of the right XIth cranial nerve was performed. The histologic analysis confirmed a diagnosis of diffuse large B-cell lymphoma. Malignant lymphoma should be considered in the differential diagnosis of pineal region tumors. Furthermore, obtaining histological confirmation is crucial for making proper management decisions.
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Yasuhiro Kiyonaga, Keisuke Maeshima, Chiharu Imada, Miwa Haranaka, Koj ...
2014 Volume 53 Issue 11 Pages
1209-1213
Published: 2014
Released on J-STAGE: June 01, 2014
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We herein report the case of an 84-year-old man with steroid-dependent adult-onset Still's disease (AOSD) whose daily steroid dose was successfully tapered after etanercept treatment. The corticosteroids worked well initially, and the patient went into remission promptly; however, he suffered a relapse due to steroid tapering. Because treatment with cyclosporine and methotrexate was ineffective, reducing the steroid dose was difficult, and the corticosteroids induced myopathy and diabetes. However, steroid tapering was accomplished in combination with etanercept therapy, and the patient's steroid-induced side effects disappeared. Etanercept should therefore be considered as a steroid-sparing treatment option in patients with steroid-responsive, steroid-dependent AOSD.
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Kazuho Morichika, Sawako Nakachi, Takeaki Tomoyose, Natsuki Shimabukur ...
2014 Volume 53 Issue 11 Pages
1215-1220
Published: 2014
Released on J-STAGE: June 01, 2014
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As a reflection of the considerable increase in the number of cancer patients treated with chemotherapy, indications for the use of implanted venous catheters are rapidly growing. However, in some cases, implanted venous catheters induce unwelcome complications. We herein report a rare case of septic pulmonary embolism (SPE) caused by local infection-associated catheter removal during the administration of ABVd combination chemotherapy consisting of adriamycin, bleomycin, vinblastine and dacarbazine in a patient with Hodgkin's lymphoma of the mixed cellularity type. During the course of treatment with chemotherapy administered via implanted venous catheters, think it is crucial to monitor for the potential occurrence of SPE.
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Keiko Yamagami, Tomoko Miyashita, Tomoyuki Nakamura, Michinori Shirano ...
2014 Volume 53 Issue 11 Pages
1221-1225
Published: 2014
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A 24-year-old man presented with fever and pleural effusion predominantly containing lymphocytes. Cultures of the pleural effusion and blood revealed
Campylobacter fetus, and laboratory studies showed a low serum level of immunoglobulin. The patient was diagnosed with
C. fetus pleuritis, bacteremia and primary hypogammaglobulinemia, and subsequent treatment with meropenem and immunoglobulin improved his condition. Although the underlying cause of the primary hypogammaglobulinemia remains unclear, the patient's status improved under immunoglobulin replacement therapy.
C. fetus pleuritis is a rare infectious disease usually observed in immunocompromised hosts. We herein describe the first report of
C. fetus pleuritis in a young adult with primary hypogammaglobulinemia.
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Chao-Ping Yu, Tian-Hu Liu, Bin Wang, Qing Zhang
2014 Volume 53 Issue 11 Pages
1227-1229
Published: 2014
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Diabetic muscle infarction (DMI) is a rare complication of long-standing diabetes mellitus. This is the first case of DMI reported by cardiologists. A 49-year-old patient with a history of diabetes and hypertension for only two years was admitted to the cardiac ward due to pain in the left thigh with pitting edema in both lower extremities. Magnetic resonance imaging finally confirmed the presence of DMI in the left thigh, which was improved by treatment with anticoagulants, analgesics and rest. However, the typical clinical symptoms of DMI were unrecognizable at the start of treatment, which may be attributed to a lack of awareness of this rare condition among non-endocrinologist physicians.
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