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Takahiko Kobayashi, Mariko Ozasa, Kencho Miyashita, Akiyoshi Saga, Kim ...
2013 Volume 52 Issue 18 Pages
2051-2056
Published: 2013
Released on J-STAGE: September 15, 2013
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Solid-pseudopapillary neoplasms (SPN) are rare pancreatic tumors. The etiology of SPN involves mutations in the gene that encodes β-catenin (
CTNNB1). We herein report the case of a 23-year-old woman with a large SPN with proliferating tumor cells that displayed both solid and pseudo-papillary patterns. The simultaneous nuclear accumulation and loss of membrane localization of β-catenin and E-cadherin was specifically observed in the tumor cells. Further, the tumor cells were shown to harbor a missense mutation in exon 3 of
CTNNB1. We also present a review of the literature describing the clustering of
CTNNB1 mutations in patients with SPN.
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Takashi Goto, Shigetoshi Ohshima, Kouichi Miura, Tomomi Shibuya, Watar ...
2013 Volume 52 Issue 18 Pages
2057-2060
Published: 2013
Released on J-STAGE: September 15, 2013
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A 70-year-old man who suffered from chronic hepatitis C was infected with HCV genotype 1 and exhibited a high viral load. He had hypertension and had consumed the equivalent of 50 g of ethanol per day. He was treated with pegylated interferon and ribavirin. After 51 weeks, he developed an unsteady gait while walking and demonstrated Barre's sign on the right foot and a headache. Contrast computed tomography showed a subdural hematoma with a mass effect. The patient was treated with drainage and aspiration surgery via a burr hole. Following the drainage procedure, there were no neurological sequelae. Treatment with pegylated interferon and ribavirin was discontinued. Fortunately, a sustained virological response was achieved.
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Kosuke Okuwaki, Mitsuhiro Kida, Hironori Masutani, Hiroshi Yamauchi, H ...
2013 Volume 52 Issue 18 Pages
2061-2066
Published: 2013
Released on J-STAGE: September 15, 2013
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Primary perivascular epithelioid cell tumors (PEComas) of the pancreas are extremely rare. We herein report our experience with a patient who had a primary PEComa of the pancreas that was diagnosed by the preoperative histopathological examination of a biopsy specimen obtained by endoscopic ultrasound-guided fine-needle aspiration (EUS-FNA). The patient was a 43-year-old woman whose chief complaint was abdominal pain. Imaging studies revealed a pancreatic tumor. Gastrointestinal stromal tumor (GIST), solid pseudopapillary tumor and neuroendocrine tumor were considered in the differential diagnosis. A histopathological examination of a specimen of the tumor obtained using EUS-FNA showed spindle-shaped tumor cells with enlarged nuclei and eosinophilic cytoplasm. The tumor cells proliferated in a sheet-like fashion and stained positive for the melanoma-associated antigen HMB-45. A PEComa was thus diagnosed. If an adequate tumor specimen can be obtained using EUS-FNA, immunostaining may facilitate the diagnosis of extremely rare diseases and therefore assist in deciding the treatment policy.
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Vito Bonomo, Mariaconcetta Di Piazza, Salvatore Evola, Ludovico La Gru ...
2013 Volume 52 Issue 18 Pages
2067-2070
Published: 2013
Released on J-STAGE: September 15, 2013
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An anomalous aortic origin of the coronary artery arising from the opposite sinus with an interarterial course is a rare condition that is associated with a high risk of sudden cardiac death during or after strenuous exertion. We herein report the case of a 47-year-old woman presenting with chest pain, syncope and palpitations who presented with a rare coronary artery anomaly on multidetector computed tomography coronary artery (MDCT-CA) with prospective electrocardiogram (ECG) gating.
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Luigi Petramala, Claudio Savoriti, Laura Zinnamosca, Cristiano Marinel ...
2013 Volume 52 Issue 18 Pages
2071-2075
Published: 2013
Released on J-STAGE: September 15, 2013
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A 25-year-old Caucasian woman was admitted to our department with severe hypokalemia that was associated with hypercalcemia. An endocrinological investigation showed the coexistence of primary hyperparathyroidism (PHPT) and primary aldosteronism (PA), arising from an adenoma of the left cortical adrenal gland. The patient underwent left laparoscopic adrenalectomy, but refused the surgical neck exploration that would be required for parathyroidectomy. The post-operative course was uneventful, and the patient realized a normalization of her potassium serum level and a reduction of her blood pressure values. We herein report the important issues regarding the management of a severe electrolyte imbalance, in view of the reciprocal interaction between aldosterone and parathyroid hormone, and their combined potential for causing cardiovascular damage.
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Akira Kurozumi, Yosuke Okada, Tadashi Arao, Itsuro Endou, Toshio Matsu ...
2013 Volume 52 Issue 18 Pages
2077-2081
Published: 2013
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Most cases of hypoparathyroidism with decreased parathyroid hormone (PTH) secretion, excluding secondary hypoparathyroidism, are considered to be idiopathic. We herein report a relatively rare case of hypoparathyroidism with extrapyramidal symptoms, including brachybasia and a frozen gait, caused by advanced basal ganglia calcification in a 64-year-old man with hypoparathyroidism. A DNA (deoxyribonucleic acid) analysis of blood samples obtained from the patient and his eldest daughter revealed autosomal dominant hypocalcemia (ADH) with mutations in the calcium-sensing receptor (CaSR) gene. In cases of chronic hypoparathyroidism, calcification of the basal ganglia is observed if the patient is not treated for a long period. However, extrapyramidal symptoms as a complication of hypoparathyroidism are relatively rare.
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Yusuke Igarashi, Tetsu Akimoto, Akinori Nukui, Hiroyasu Nakamura, Akir ...
2013 Volume 52 Issue 18 Pages
2083-2086
Published: 2013
Released on J-STAGE: September 15, 2013
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Decreases in blood pressure develop in response to a wide range of clinical disorders. Various factors have been implicated in the development of hemodialysis-associated hypotension, including an impairment of the compensatory processes, an autonomic dysfunction or cardiac failure. The additional presence of concomitant acute abdomen may result in a diagnostically challenging situation. We herein report our experience with a chronic hemodialysis patient who developed severe hypotension with acute flank pain due to spontaneous bleeding around the kidney. Concerns regarding the management of the disease are also discussed.
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Ayako Kokuzawa, Yoshiyuki Morishita, Hiromichi Yoshizawa, Kana Iwazu, ...
2013 Volume 52 Issue 18 Pages
2087-2091
Published: 2013
Released on J-STAGE: September 15, 2013
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We herein report the case of a 17-year-old man who developed an increased plasma creatinine level (11.1 mg/dL) and oliguria with massive proteinuria (27.3 g/day) four weeks after an abraded wound to his right knee. The histology of the renal biopsy specimens showed diffuse endocapillary proliferative glomerulonephritis with the deposition of nephritis-associated plasmin receptor in the glomerulus. A case of acute kidney injury due to nephrotic syndrome caused by acute post-streptococcal glomerulonephritis was diagnosed. His renal function and proteinuria were improved with supportive care, including hemodialysis, without the administration of immunosuppressive agents.
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Yuko Nakashima, Kenji Ito, Hitoshi Nakashima, Aki Shirakawa, Yasuhiro ...
2013 Volume 52 Issue 18 Pages
2093-2097
Published: 2013
Released on J-STAGE: September 15, 2013
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A 43-year-old man was admitted with end-stage renal disease caused by IgA nephropathy, and was treated with maintenance peritoneal dialysis. The patient developed general fatigue and appetite loss, and his symptoms were gradually aggravated by depression. After approximately 2 months on dialysis, the patient presented with altered consciousness and ophthalmoplegia. Wernicke's encephalopathy was diagnosed based on the presence of classic symptoms and the findings on magnetic resonance imaging. Thiamine replacement therapy was immediately initiated. The patient recovered from most of his neurological symptoms; however, the sequela of Korsakoff syndrome remained. A marginal thiamine deficiency in combination with predisposing factors must be considered when treating dialysis patients.
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Tomotsune Miyamoto, Yozo Ishikawa, Junya Yamamoto, Tsuyoshi Yamamura, ...
2013 Volume 52 Issue 18 Pages
2099-2103
Published: 2013
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A 79-year-old woman with familial hyperlipidemia was treated with low-density lipoprotein apheresis. She was hospitalized due to fatigue and edema, and massive proteinuria was discovered. Renal biopsy revealed no distinct abnormalities, thus suggesting a diagnosis of minimal change nephrotic syndrome. She developed acute kidney injury and hemodialysis was initiated. Two series of steroid pulse therapy were given, but the proteinuria did not decrease. Thereafter, she developed thrombocytopenia and fell into a stupor. Thrombotic microangiopathy (TMA) was the most likely diagnosis. Plasma exchange was initiated, resulting in improvements in platelet counts and in her level of consciousness. Clinicians should therefore be aware that TMA can occur as a result of steroid pulse therapy.
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Hiroki Suzuki, Mari Watanabe, Tomoka Ara, Midori Nishizuka, Mami Morit ...
2013 Volume 52 Issue 18 Pages
2105-2111
Published: 2013
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The full picture of immunoglobulin G4-related lung disease (IgG4-RLD) has not yet been elucidated. A 69-year-old man was referred to us with a more than 2-week history of productive cough and fatigue. Chest CT showed an airspace consolidation along the bronchovascular bundles. The pathological findings that were obtained from an open-lung biopsy showed both organizing pneumonia and interstitial pneumonia. Based on the established, comprehensive diagnostic criteria for IgG4-related disease (RD) as of 2011, this patient was given a definitive diagnosis of IgG4-RD. A further accumulation and analysis of those cases that concomitantly present with both IgG4-RLD and organizing pneumonia, like our patient, may contribute to the elucidation of the pathology of IgG4-RLD and the establishment of the disease spectrum.
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Chan Woo Park, Woojin Kim, In Jae Oh, Kyu Sik Kim, Yoo Duk Choi, Yong ...
2013 Volume 52 Issue 18 Pages
2113-2116
Published: 2013
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Solitary extramedullary plasmacytoma (SEP) is a plasma cell neoplasm that develops outside of the bone marrow. A solitary, exophytic growth in the airway is an extremely rare presentation of SEP. We herein report a case of SEP presenting as an endobronchial mass. The tumor was treated by rigid bronchoscopic debulking followed by ablation using argon plasma coagulation. However, the tumor could not be completely removed due to its wide base. Adjuvant radiotherapy was administered as the curative therapy. A biopsy was performed on the resected specimen and the diagnosis of plasmacytoma was thereby confirmed.
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Hideaki Yamakawa, Noboru Takayanagi, Takashi Ishiguro, Yosuke Miyahara ...
2013 Volume 52 Issue 18 Pages
2117-2119
Published: 2013
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A 38-year-old man with Marfan syndrome underwent an aortic replacement with an artificial aortic valve at 27 years of age and an aortic graft at 31 years of age. In 2011, he was diagnosed as having chronic necrotizing pulmonary aspergillosis (CNPA). He developed a fever and an increased sputum volume and was admitted to the hospital in 2012. Contrast-enhanced CT showed an irregularly shaped nonenhanced structure in the aortic graft. He died on hospital day 31. From the autopsy findings, we speculated that an infiltration by the CNPA lesion into a pulmonary vein was followed by the hematogenous formation of a fungus ball in the aortic graft.
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Atsushi Iwata, Kenji Ishihara, Shoji Tsuji
2013 Volume 52 Issue 18 Pages
2121-2122
Published: 2013
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We herein report a case of late-onset Huntington's disease (HD) that presented without any involuntary movement. The patient was in her forties at onset, and she lacked any rigidity; thus, the HD in this case was completely different than the Westphal variant. The diagnosis was made by confirming the expansion of CAG repeats in the HD gene after obtaining information about her sister's clinical features, since her sister presented with typical HD. In late onset HD, involuntary movement is thought to be the core feature; therefore, the clinical features of this case are considered to be unique and noteworthy.
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Takashi Nomura, Shinya Kawase, Yasuhiro Watanabe, Kenji Nakashima
2013 Volume 52 Issue 18 Pages
2123-2126
Published: 2013
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We used ramelteon to treat two patients with secondary REM sleep behavior disorder (RBD) complications along with neurodegenerative diseases including multiple system atrophy and Parkinson's disease. These two patients not only improved in terms of their clinical RBD symptoms but also exhibited a decrease in the proportion of REM sleep without atonia (from 8.5% to 3.5% and from 10.9% to 3.9% in the two patients). Although clonazepam is the standard first-line therapy for the treatment of RBD, ramelteon might be an effective treatment alternative in patients with RBD who cannot take clonazepam due to either ineffectiveness or adverse effects.
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Kosuke Matsuzono, Takao Yoshiki, Yosuke Wakutani, Yasuhiro Manabe, Tor ...
2013 Volume 52 Issue 18 Pages
2127-2129
Published: 2013
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We herein report a 53-year-old female with repeated transient ischemic attack (TIA) symptoms including 13 instances of right hemiparesis that decreased in duration over 4 days. Two separate examinations using diffusion weighted image (DWI) in magnetic resonance imaging (MRI) revealed normal findings, but we observed that both Babinski and Chaddock signs were completely synchronized with her right hemiparesis. We were only able to diagnose this case of early stage TIA using clinical signs. This diagnosis was confirmed 4 days after the onset by the presence of abnormalities on the MRI. DWI-MRI is generally useful when diagnosing TIA, but a neurological examination may be more sensitive, especially in the early stages.
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Francesco Tovoli, Antonio Vannini, Chiara Masi, Tiziana Balbi, Luigi B ...
2013 Volume 52 Issue 18 Pages
2131-2134
Published: 2013
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A 23-year-old man was referred to our Emergency Department due to an acute-onset hemoptysis and the associated bilateral swelling of all of his major salivary glands. The elevated levels of systemic inflammatory markers with hypereosinophilia and the concurrent presence of multiple lung infiltrates made it difficult to perform a differential diagnosis, as these symptoms can also be indicative of infectious, autoimmune or hematologic disorders. A histological examination of the patient's left submandibular gland revealed strong clues as to the final diagnosis of eosinophilic granulomatosis with polyangiitis with an atypical clinical presentation, thus allowing for the administration of early and successful conservative therapy. The outcome of our case suggests that systemic vasculitis represents a rare but possible cause of acute bilateral sialadenitis in young patients.
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Yoshiko Yoshida, Toshihiro Shirai, Masashi Mikamo, Yuichiro Shishido, ...
2013 Volume 52 Issue 18 Pages
2135-2138
Published: 2013
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A 55-year-old woman visited our hospital for an investigation of central bronchiectasis, mucoid impaction and infiltrative shadows on chest CT. She had a 10-year history of bronchial asthma; however, her adherence to treatment was poor. Based on the presence of peripheral blood eosinophilia and immediate cutaneous reactivity to
Aspergillus fumigatus, the patient was clinically diagnosed with allergic bronchopulmonary aspergillosis. Her condition and CT findings improved with systemic corticosteroid therapy. It was found that the patient had not been sensitized to
Aspergillus 10 years earlier, indicating that single testing is inadequate for the early diagnosis of this disease.
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Hideharu Hagiya, Hiroki Kajioka
2013 Volume 52 Issue 18 Pages
2139-2143
Published: 2013
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We herein report the case of an 85-year-old woman presenting with right internal jugular vein candidal thrombophlebitis associated with central venous catheters (CTCVC). The infecting agent was
Candida albicans, which caused recurrent candidemia five times in total. Micafungin (MCFG) alone was ineffective; however, the combination of MCFG with fosfluconazole (F-FLCZ) successfully treated the patient without a need for any anticoagulant or surgical therapies. To the extent of our knowledge, this is the first report of CTCVC being successfully treated with a combination of F-FLCZ and MCFG. These new antifungal agents have better efficacy, tolerability and bioavailability; therefore, they can be useful alternatives to classical combination therapies such as amphotericin-B and 5-fluorocytosine.
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Toshimasa Hayashi, Hideki Uchiumi, Kunio Yanagisawa, Yoshiyuki Ogawa, ...
2013 Volume 52 Issue 18 Pages
2145-2147
Published: 2013
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Systemic infection caused by
G. haemolysans has rarely been reported. We herein describe the case of a 69-year-old woman with recurrent
G. haemolysans meningitis that led to abducens nerve palsy. Osteomyelitis of the clivus was likely present at the first admission, which led to reinfection of the meninges because the course of antibiotic treatment was too short. The patient has remained free of relapse for one year after undergoing a second round of treatment that lasted 63 days. In cases of
G. haemolysans meningitis, coexisting infectious diseases, such as endocarditis and/or osteomyelitis, should therefore be investigated to prevent recurrence.
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Mitsuru Sada, Takeshi Saraya, Yasutaka Tanaka, Shinji Sato, Megumi Wak ...
2013 Volume 52 Issue 18 Pages
2149-2153
Published: 2013
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A 45-year-old man was referred to our hospital with a 3-month history of dyspnea, polyarthralgia, myalgia and weight loss. He was diagnosed with systemic lupus erythematosus/dermatomyositis overlap syndrome with lung involvement, which presented as organizing pneumonia. However, a bronchoscopic examination revealed the presence of multiple plaque-like white lesions with ulcers on the bronchial membrane, located mainly in the central airway. The pathological specimens obtained from bronchoscopy showed numerous filamentous fungal hyphae that were aggressively invading the bronchial walls, suggesting a diagnosis of invasive tracheobronchial aspergillosis. The present case, along with a review of the literature, demonstrates that invasive tracheobronchial aspergillosis can occur in patients who do not appear to be immunosuppressed. This case of aspergillosis should thus be recognized as an extremely rare presentation of an
Aspergillus infection.
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Kenta Hachiya, Tomonori Sugiura, Yasuaki Dohi, Nobuyuki Ohte
2013 Volume 52 Issue 18 Pages
2155-2156
Published: 2013
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Yukiharu Sugimura, Masanori Katoh, Masaaki Toyama
2013 Volume 52 Issue 18 Pages
2157-2158
Published: 2013
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Toshio Kahara, Tomomi Ichikawa, Hirokazu Taniguchi, Hideki Shinnou, Hi ...
2013 Volume 52 Issue 18 Pages
2159
Published: 2013
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Jui-Hsiang Lin, Ja-Liang Lin, Chiao-Yang Lu, Tzung-Hai Yen
2013 Volume 52 Issue 18 Pages
2161
Published: 2013
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Yosuke Miyaji, Takashi Kurokawa, Fumiaki Tanaka, Kazuo Koyama
2013 Volume 52 Issue 18 Pages
2163-2164
Published: 2013
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Megumi Toko, Hiroshi Oiwa, Shoji Mihara, Eiji Sugiyama
2013 Volume 52 Issue 18 Pages
2165
Published: 2013
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Shih-Horng Huang, Chih-Cheng Lai, Sheng-Hsiang Lin
2013 Volume 52 Issue 18 Pages
2167
Published: 2013
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