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Since its clinical definition in 1987, the syndrome called, "periodic fever, aphtous stomatitis, pharyngitis and cervical adenitis" syndrome (PFAPA) has been considered peculiar to pediatric age. In the recent literature there are a few case reports of PFAPA in adults. We describe a case of a 21-year-old female affected by PFAPA who presented a history of tonsillectomy at the age of four. To our knowledge this is the fourth case described with a diagnosis of PFAPA in an adult with a history of tonsillectomy during childhood. Although the role of tonsillectomy in the treatment of PFAPA is still controversial, due to the lack of definitive data in literature, this case suggests that fever episodes may relapse several years after surgery.
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Patients enrolled in the mepolizumab trial were evaluated for L-HES on the basis of T-cell phenotyping and T-cell receptor gene rearrangement patterns, and their serum thymus-and-activation-regulated chemokine (TARC) levels were measured. Response to treatment was compared in patient subgroups based on results of these analyses.
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We describe a case of a primary central nervous system ILPD in a patient with ulcerative colitis treated chronically with the immunomodulatory agents infliximab and azathioprine. This 52-year-old woman presented with a 1-month history of left-side weakness and paresthesias. Neuroimaging identified multiple heterogeneously enhancing lesions in her cerebrum. Extensive systemic infectious and malignancy-related investigations were negative, prompting neurosurgical referral to obtain a tissue diagnosis. Pathologic assessment of her open excisional biopsy specimen confirmed the diagnosis of a polymorphic lymphoproliferative disorder. She was treated by withdrawal of infliximab and azathioprine, along with a prolonged course of prednisone. At her 6-month follow-up, she demonstrated both clinical and radiologic improvement.
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A phase 3 trial assessing response-based therapy in intermediate-risk Hodgkin lymphoma mandated real-time central review of involved field radiation therapy (IFRT) and imaging records by a centralized review center to maximize protocol compliance. We report the impact of centralized radiation therapy review on protocol compliance.
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A 63-year-old Caucasian male with history of hypertension was treated for pulmonary sarcoidosis with a 6-month course of prednisone. His creatinine was 1.6 mg/dL during the course. Two months after finishing treatment, he presented with creatinine of 4 mg/dL. A kidney biopsy was performed, which showed nonspecific changes without evidence of granuloma or active interstitial inflammation. He was empirically started on prednisone for presumed renal sarcoidosis, even with a nondiagnostic kidney biopsy finding. Within a month of treatment, his serum creatinine improved to 2 mg/dL, though not to baseline. He continues to be stable on low-dose prednisone. With this case as a background, we aimed to determine the incidence of inconclusive kidney biopsies in patients with sarcoidosis presenting with AKI and to identify the various histological findings seen in this group of patients.
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PF was induced in mice by intratracheal instillation of bleomycin (5 mg/kg). The mice were orally treated with paeoniflorin or prednisone for 21 d. After the mice were sacrificed, lung tissues were collected for analysis. An in vitro EMT model was established in alveolar epithelial cells (A549 cells) incubated with TGF-β1 (2 ng/mL). EMT identification and the expression of related proteins were performed using immunohistochemistry, transwell assay, ELISA, Western blot and RT-qPCR.
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Clinical features and treatment outcomes of intravascular large B-cell lymphoma (IVLBCL) have rarely been reviewed due to its rarity and pathologic obscurity.
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To identify the underlying mechanism of amenorrhea in juvenile systemic lupus erythematosus (JSLE) patients, thirty-five (11.7%) JSLE patients with current or previous amenorrhea were consecutively selected among the 298 post-menarche patients followed in 12 Brazilian pediatric rheumatology centers. Pituitary gonadotrophins [follicle-stimulating hormone (FSH) and luteinizing hormone (LH)] and estradiol were evaluated in 32/35 patients, and prolactin and total testosterone in 29/35 patients. Patient's medical records were carefully reviewed according to demographic, clinical and therapeutic findings. The mean duration of amenorrhea was 7.2 ± 3.6 months. Low FSH or LH was observed in 7/32 (22%) JSLE patients and normal FSH or LH in 25 (78%). Remarkably, low levels of FSH or LH were associated with higher frequency of current amenorrhea (57% vs. 0%, P = 0.001), higher median disease activity (SLEDAI) and damage (SLICC/ACR-DI) (18 vs. 4, P = 0.011; 2 vs. 0, P = 0.037, respectively) and higher median current dose of prednisone (60 vs. 10 mg/day, P = 0.0001) compared to normal FSH or LH JSLE patients. None of them had decreased ovarian reserve and premature ovarian failure. Six of 29 (21%) patients had high levels of prolactin, and none had current amenorrhea. No correlations were observed between levels of prolactin and SLEDAI, and levels of prolactin and SLICC/ACR-DI scores (Spearman's coefficient). We have identified that amenorrhea in JSLE is associated with high dose of corticosteroids indicated for active disease due to hypothalamic-pituitary-ovary axis suppression.
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Myofibroblastic tumors are inflammatory tumors that arise in viscera and soft tissue; their etiopathology is poorly understood. They are capable of infiltration of adjacent organs, local recurrence after surgical resection, and even of distant metastasis. These characteristics result in persistent debate as to the nature of these lesions - whether they are inflammatory or neoplastic, benign or malignant lesions? Diagnosis is almost always made based on histopathological findings. Traditional management is complete surgical excision, but this may be difficult or impossible when the lesion develops in proximity to vital structures. We report the case of a 59-year-old man who was treated in our institution for mesenteric myofibroblastic tumor. Complete resection was not possible due to local infiltration of the mesentery. Recurrence was noted 3 months after surgery; treatment with steroidal and then non-steroidal anti-inflammatory medications (NSAIDs) resulted in clinical and radiologic regression of the tumor.
Calcium absorption measured by dual isotope absorptiometry was elevated at 37.4%. Serum levels of 24,25-dihydroxyvitamin D were very low, as measured in two laboratories (0.62 ng/mL [normal, 3.49 ± 1.57], and 0.18 mg/mL). Genetic analysis of CYP24A1 revealed homozygous mutation E143del previously described. The patient's serum calcium and renal function improved markedly on treatment with ketoconazole but not with prednisone.
PredCH is designed to assess whether oral prednisone added to first-line agent verapamil helps reduce the number and intensity of cluster attacks in the beginning of a cluster episode as compared to monotherapy with verapamil.
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The authors present an unusual case of neurosarcoidosis masquerading as a CNS glioma. In cases of solitary CNS granulomas, radiographically differentiating neurosarcoidosis from a glioma can be challenging. In this case, serial ophthalmic examination identifying sequential involvement of both optic nerves helped to identify the underlying cause of the CNS disease as sarcoidosis.
These results suggest that Tregs may contribute to the immunopathogenesis of OLP and may provide a new therapeutic target for OLP treatment.
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Leukocytoclastic vasculitis (LV) is a rare hypersensitive reaction involving the small vessels, which is usually mediated by drugs. Very few cases of nafcillin -associated LV have been reported. Here, we reported a case of LV with the presentation of skin rashes and gastrointestinal bleeding after receiving nafcillin, evidenced by endoscopy and skin biopsy. The symptoms resolved after withdrawal of nafcillin and the addition of prednisone treatment. LV should be considered in the differential diagnosis of erythematous rash, especially with gastrointestinal symptoms after the exposure.
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Discussion of a rare case of angioleiomyoma of the internal auditory canal.
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IgG4-related disease and MPO-ANCA-associated vasculitis limited to the central nervous system can account for a high percentage of the cases of hypertrophic pachymeningitis that were considered idiopathic, and their diagnosis requires a biopsy and a histological study.
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To analyze survival time and identify prognostic factors associated with outcome following discharge in dogs with primary brain tumors treated palliatively.
Because of prednisone-related suppression of IFN-score, higher accuracy of rituximab response prediction was achieved in PREDN(-) patients. These results suggest that the IFN-score-based rituximab response prediction model could be improved upon implementation of prednisone use.
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We consider a conceptual correspondence between the missing data setting, and joint modeling of longitudinal and time-to-event outcomes. Based on this, we formulate an extended shared random effects joint model. Based on this, we provide a characterization of missing at random, which is in line with that in the missing data setting. The ideas are illustrated using data from a study on liver cirrhosis, contrasting the new framework with conventional joint models.
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Simultaneous tumor necrosis factor (TNF)-α levels were significantly increased. The patient had a dramatic recovery that was confirmed by a repeated computed tomography scan, and the levels of TNF-α returned to normal after 2 weeks of methylprednisolone treatment.
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Rituximab in combination with chemotherapy improves CR rate, DFS and OS. Further prospective trials are needed to confirm our results.
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Corticosteroid doses and the length of treatment were not recorded in the NAMCS data.
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Kasabach-Merritt syndrome (KMS) refers to the combination of large neonatal vascular tumors and thrombocytopenic coagulopathy. However, a standard treatment regimen for KMS has not yet been established. We report a case of a 6-week-old male infant with life-threatening KMS who was successfully treated with transarterial embolization and corticosteroids. One week after initiating the corticosteroid treatment, his platelet counts recovered, and the lesion growth halted. The approach with corticosteroid therapy resulted in an excellent response that was maintained long enough for us to perform transarterial embolization therapy. The combination of transarterial embolization and corticosteroid therapy should be considered as an option for Kasabach-Merritt syndrome.
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Early and maintained targeted treatment has functional and radiological benefits over the first 7 years of the BeSt study and can include drug tapering and (partial, temporary or permanent) discontinuation.
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A boy aged 5 years 8 months presented with relapsing optic neuritis, short segment transverse myelitis, and brain involvement. He met the diagnostic criteria for multiple sclerosis fulfilling the McDonald 2010 criteria; however, neuromyelitis optica immunoglobulin-G was detected, and the patient was diagnosed with neuromyelitis optica. He had frequent relapses until immunosuppressive treatment with azathioprine and low-dose prednisone was started. After he was asymptomatic for 2.5 years, prednisone was withdrawn, but he had a new attack soon after withdrawal of the steroid.