Objective To analyze the clinical characteristics of patients with gastrointestinal bezoars and their response to therapy. Patients We retrospectively reviewed the cases of 19 patients diagnosed with gastrointestinal bezoars at the Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical Sciences or one of 15 collaborating institutions between December 2004 and August 2013. We investigated the epidemiology and etiology of the gastrointestinal bezoars by determining the gender, age at diagnosis, medical history, symptoms, complications, modalities used for diagnosis, treatments, outcomes and bezoar location, color and contents. Results There were 17 patients with gastric bezoars and two patients with small intestinal bezoars. All patients were 62 years of age or older, except for one case of a trichobezoar in a 10-year-old patient. Some of the patients had a history of surgery of any part of the gastrointestinal tract (n=5) and/or diabetes mellitus (n=2). The two patients with small intestinal bezoars required surgical removal in order to relieve ileus. Approximately one-half of the patients with gastric bezoars had ulcerations in the stomach (9/17 patients, 52.9%) and/or gastrointestinal bleeding (8/17, 47.1%). Endoscopic fragmentation was performed in 10 patients, whereas bezoar dissolution was achieved with a gastroprokinetic agent (n=1) and without any treatment (n=3) in the remaining cases. Conclusion As previously reported, elderly individuals with a positive history of surgery and/or diabetes mellitus were observed in this bezoar patient series. Gastric ulcers and gastrointestinal bleeding were frequently observed. The majority of patients underwent endoscopic fragmentation, while spontaneous resolution of the gastric bezoar was observed in several cases.
Objective The Japanese health insurance system approved the use of proton pump inhibitors (PPIs) for the prevention of peptic ulcers in patients using low-dose aspirin (LDA) and/or non-steroidal anti-inflammatory drugs (NSAIDs). However, many orthopedists and physicians do not prescribe PPIs to elderly patients with atrophic gastritis. The aim of this study was to determine whether PPIs are effective in preventing gastrointestinal mucosal injury in elderly Japanese patients with atrophic gastritis. Methods We examined the associations between the use of antiulcer drugs and endoscopic findings in elderly Japanese patients using LDA or NSAIDs. Patients We evaluated 100 patients using LDA and 58 patients using non-aspirin NSAIDs 65 years of age or older. All patients underwent upper GI endoscopy to detect the presence of open ulcers and hemorrhagic lesions and assess the extent of atrophic gastritis. Results Among the patients using LDA, the prevalence of open ulcers was significantly lower in the patients using PPIs than in those using mucosal protective agent only and those not receiving antiulcer treatment (p<0.001). Among the patients using NSAIDs, the patients treated with PPIs exhibited a significantly lower incidence of open ulcers than the patients not receiving antiulcer treatment (p=0.012). Open-type atrophic gastritis was observed in nearly 70% of the patients. Conclusion The use of PPI treatment is advisable in order to prevent the discontinuation of LDA or NSAIDs due to the development of gastrointestinal disorders in elderly patients with atrophic gastritis.
Objective Non-HIV patients with pneumocystis pneumonia (PCP) have a poor prognosis. We aimed to evaluate the prognostic factors for in-hospital mortality in terms of the clinical findings, including the results of bronchoalveolar lavage fluid (BALF)-analyses, in non-HIV PCP patients. Methods We retrospectively reviewed non-HIV PCP patients diagnosed using bronchoalveolar lavage between April 2006 and July 2012. For patients with a poor respiratory status, noninvasive positive pressure ventilation (NPPV) was used during the bronchoalveolar lavage (BAL) procedure. Data regarding demographics, laboratory findings and the prognosis were evaluated. Results A total of 29 non-HIV PCP patients were analyzed. NPPV was carried out safely and successfully in 12 patients during the BAL procedure. Twelve patients (41%) died. The multivariate logistic regression analysis identified only BALF neutrophilia to be a significant prognostic factor determining in-hospital mortality. The log-rank test showed that the patients with BALF neutrophilia (≥31%) had a significantly lower survival rate than the other patients (p=0.001). Conclusion Only BALF neutrophilia was found to be a significant predictor of survival in patients with non-HIV PCP. Our data also emphasize the significance of performing BAL in such patients, as it provides both diagnostic and prognostic information.
Objective To evaluate the performance and practicality of QuantiFERON TB-2G (QFT-2G) testing for screening healthcare workers (HCWs) at a city hospital in Japan without a tuberculosis (TB)-specific ward. Methods We performed a chart review of 951 HCWs (251 men and 700 women) who underwent QFT-2G testing as a part of their pre-employment or annual employee screening between April 2007 and March 2010. Results The initial QFT-2G test was interpreted as positive in 28 (2.9%) HCWs, negative in 884 HCWs (92.9%) and indeterminate in 39 HCWs (4.1%). During the four-year study period, 37 HCWs were diagnosed as being positive at least once. Nine (0.98%) of the 923 HCWs with indeterminate or negative results on the initial testing converted to a positive status, including 6/479 (1.25%) nurses, 2/100 (2.0%) office staff members and 1/147 (0.68%) physicians. No HCWs with a positive result had a history of tuberculosis (TB) or any apparent contact with active TB patients and did not opt for treatment of latent TB. Seven (25%) of the 28 HCWs who were determined to be positive on the initial testing reverted to an indeterminate or negative status. Conclusion In a series of annual serial QFT-2G tests, some HCWs exhibited conversion and/or reversion. Therefore, caution is required when interpreting mild fluctuations in interferon-γ responses.
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.
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.
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 PaO2 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.
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.
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.
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.
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.
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/m2/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.
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.
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.
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.
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.
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.
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.
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.
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.
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.
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.
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.
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.