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PubMed2006

Effectiveness of steroid treatment for hoarseness caused by idiopathic fibrosing mediastinitis: report of a case.

A 65-year-old woman was referred to our department for investigation and treatment of hoarseness. A chest computed tomography (CT) scan showed a mediastinal mass spreading into the aortopulmonary window. This finding and that of flexible laryngoscopy suggested that the hoarseness was being caused by left recurrent nerve involvement resulting in left vocal cord paralysis. Thus, we performed a mediastinoscopic biopsy via a parasternal incision. Pathological examination revealed dense fibrous tissue infiltrated with varied inflammatory cells. Because no etiological pathogen or neoplastic lesion was identified, we diagnosed idiopathic fibrosing mediastinitis and began treating the patient with prednisolone. After a course of treatment, the mediastinal lesion showed a remarkable response. The hoarseness resolved as the lesion became smaller. Laryngoscopy confirmed recuperation of vocal cord function. This report shows that steroid therapy is a treatment option for hoarseness caused by recurrent laryngeal nerve involvement of fibrosing mediastinitis, if administered under close observation.

Aged↗
PubMed1986

The clinical syndrome of fibrositis.

Fibrositis is a disorder of musculoskeletal pain and aching with at least a five to one female-to-male ratio. It is most commonly seen between the ages of 40 and 60, and has a prevalence in the clinic of 6 to 15 percent. Its most common mode of presentation involves generalized musculoskeletal pain and aching, but articular pain, axial skeletal pain, myalgias, and neurovascular complaints sometimes predominate. All patients have multiple areas of local tenderness called "tender points" that are easily identified during physical examination, and are diagnostic. Essential symptoms of fibrositis are disturbed sleep, morning stiffness, and fatigue. Additional rheumatic symptoms include subjective swelling, paresthesias, and numbness. Headaches and irritable bowel syndrome are common nonrheumatic complaints. Modest improvement follows treatment by tricyclic agents such as low-dose cyclobenzaprine and amitriptyline, by physical measures, and by reduction in stress. Remission occurs in 20 percent of patients, but is generally short-lived.

Adult↗
PubMed1986

Pharmacotherapeutics in fibrositis.

Fibrositis (fibromyalgia) is best treated by attaining patient acceptance of and compliance with a comprehensive treatment program. This includes education, physical therapy, muscle and mental relaxation, examination and adjustment of goals and priorities, and pacing of activities, as well as pharmacotherapeutics. Of the medications in use, anti-inflammatory agents with a high degree of analgesia are more effective than "pure" anti-inflammatory agents. Tricyclic derivatives are beneficial muscle relaxants. The use of long-acting agents improves compliance and is therefore favored. Local agents are of adjunctive value in some cases. Double-blind, placebo-controlled studies show cyclobenzaprine to be an effective medication in the treatment of fibrositis (fibromyalgia).

Amitriptyline↗
PubMed1992

Fibrosing cytolytic liver failure secondary to recurrent hepatitis B after liver transplantation.

Four patients who underwent transplantation for hepatitis B virus-related liver disease developed rapidly progressive liver failure attributable to recurrent hepatitis B disease typified by hyperbilirubinemia and distinctive hepatocyte ballooning and progressive fibrosis consistent with recently reported fibrosing cholestatic hepatitis. Among these four patients, the mean interval from transplantation to redocumentation of hepatitis B surface antigen (HBsAg) and hepatitis B e antigen (HBeAg) was 5 months, to development of malaise and jaundice 6 months, to histological diagnosis 7 months, and to graft failure 8 months. The only patient who underwent retransplantation had accelerated recurrence of the same syndrome with biopsy documentation 1 month later and graft failure 2 months later. Distinctive histological features included confluent hepatocellular ballooning and progressive periportal fibrosis followed by lobular collapse over 4-6 weeks without significant inflammation. Immunohistochemical staining showed marked HBsAg and hepatitis B core antigen (HBcAg) immunoreactivity. The rapid development of cytolytic hepatocellular necrosis and lobular collapse with prominent HBcAg immunoreactivity without significant inflammation suggests a cytolytic rather than immune pathogenesis for this unique and devastating form of recurrent hepatitis B that might better be termed "fibrosing cytolytic hepatitis."

Adult↗
PubMed1994

Fibrosing cholestatic hepatitis in a hepatitis B surface antigen carrier after renal transplantation.

A 45-year-old hepatitis B surface antigen carrier had an allograft kidney transplantation and maintenance immunosuppression with cyclosporin A and prednisolone. Six months later, she experienced a rapidly progressive hepatic failure manifested by elevation of serum bilirubin level, prolongation of prothrombin time, and mild to modest increase of serum aminotransferase levels. She died in 6 weeks. Postmortem liver histology showed canalicular and cellular cholestasis and ground-glass appearance and ballooning of most hepatocytes, but only mild inflammatory cell infiltration. Immunohistochemical staining showed massive loads of hepatitis B surface and core antigens in the hepatocytes and extensive periportal fibrosis. The whole picture was compatible with fibrosing cholestatic hepatitis described in hepatitis B virus-infected liver transplant. Sequencing of the hepatitis B virus genome amplified from the patient's serum indicated a precore mutant but few mutations in the core, pre-S, and S genes. Little inflammatory reaction was observed histologically despite HLA compatibility, a situation differing from that in liver transplant. This observation indicates that fibrosing cholestatic hepatitis may also occur in non-liver transplant setting.

Base Sequence↗
PubMed1992

A thyroid biopsy with histologic features of both Riedel's thyroiditis and the fibrosing variant of Hashimoto's thyroiditis.

We describe a 36-year-old woman with clinical, laboratory, and histologic features of both Riedel's thyroiditis and the fibrosing variant of Hashimoto's thyroiditis. Features of the former included a hard, fixed thyroid mass and extensive involvement of perithyroidal tissues by dense fibrosis with lymphocytes, histiocytes, and plasma cells. Features supporting Hashimoto's thyroiditis included high serum titers of antimicrosomal and antithyroglobulin antibodies and the histologic findings within the thyroid gland itself: dense fibrous bands dividing the thyroid parenchyma into nodules composed of lymphoid follicles with germinal centers, plasma cells, and oxyphilic metaplasia of follicular epithelial cells. Although Riedel's thyroiditis and the fibrosing variant of Hashimoto's thyroiditis were once considered morphologic variants of the same disease, since the 1970s these diseases have been considered as distinct clinicopathologic entities. The coexistence of both diseases in a patient is rare and is probably coincidental in this instance.

Adult↗
PubMed1995

Fibrosing cholestatic hepatitis in a renal transplant recipient associated with the hepatitis B virus precore mutant.

A patient with evidence of chronic infection with hepatitis B virus (seropositive for hepatitis B surface antigen, and with antibody to hepatitis B e antigen) underwent renal transplantation, which subsequently failed. The patient developed abnormal liver function tests and 8 months after the removal of the transplanted kidney, hepatitis B virus DNA was detected in the serum in the absence of hepatitis B e antigen. Liver biopsy revealed the presence of fibrosing cholestatic hepatitis. Sequence analysis of the serum hepatitis B virus DNA showed the presence of the pre-core mutant. This case shows that fibrosing cholestatic hepatitis can occur outside the setting of liver transplantation, and coincided with the development of the pre-core mutant of hepatitis B virus.

Base Sequence↗
PubMed1991

Chronic fibrosing pancreatitis in a 12-year-old female.

Recurrent abdominal pain in an adolescent population is a frequent complaint. However, diseases of the pancreas, and especially chronic pancreatitis, in this age group are extremely uncommon. One type of pancreatitis, fibrosing pancreatitis, has been reported in only 14 previous pediatric patients, five of whom were over 12 years of age. We report an additional 12-year-old female with this condition. This case serves to remind us that fibrosing pancreatitis needs to be considered in any adolescent patient with chronic abdominal pain, recurrent vomiting, weight loss, steatorrhea, and painless obstructive jaundice.

Abdominal Pain↗
PubMed1991

Unattainable homeostasis in healing leads to interstitial and vascular intimal fibroses: a unified hypothesis of their pathogenesis.

Homeostasis, or the maintenance of constant internal environment in the organism, is disrupted by injury. In this case, pathological defence reactions such as inflammation and healing take place in order to restore it. They succeed if etiological harmful factors are eliminated. In the opposite case, constant injury leads to continuous healing and fibrosis. Both interstitial and vascular intimal fibroses have similar pathogeneses and may be provoked by the same etiological factors. It may be concluded, therefore, that in spite of their apparent differences, both fibroses are only the different expressions of the same process by which injury should be healed and homeostasis restored. Fibrosis itself may become dangerous for the patient, depending on its extent and on the affected organ. In this case, the inhibition of healing reaction by antiangiogenesis may be envisaged as a life-saving measure.

Animals↗
PubMed1993

One-stage allotransplantation of thoracic segment of the trachea in a patient with idiopathic fibrosing mediastinitis and marked tracheal stenosis.

The authors observed a 24-year-old female with idiopathic fibrosing mediastinitis affecting the thoracic segment of the trachea and producing marked stenosis. Taking into consideration the common occurrence of cicatricial stenosis and the inefficacy of its conservative treatment, an attempt at one-stage allotransplantation of the trachea with omentopexy of the graft and the lines of anastomosis was made. In spite of immunosuppressive therapy (cyclosporine and azathioprine), signs of rejection appeared on the 10th postoperative day, which were arrested by antithymocyte globulin and loading doses of corticosteroids. Subsequently, the condition of the patient became stable. By the end of the 2nd month the graft appeared vital, its lumen being about 10 mm, the lines of anastomoses were epithelialized. Four months after operation signs of the graft stenosis appeared, possibly caused by progressive fibrosing mediastinitis. Because of this complication a silicon stent was used for the prophylaxis of further graft stenosis. This satisfactory result of tracheal allotransplantation is thought to be related to adequate selection of the donor-recipient pair, modern immunosuppressive therapy and utilization of omentopexy for early graft revascularization.

Adult↗
PubMed2006

Nephrogenic fibrosing dermopathy/nephrogenic systemic fibrosis with diaphragmatic involvement in a patient with respiratory failure.

Nephrogenic fibrosing dermopathy/nephrogenic systemic fibrosis (NFD/NSF) is a disorder occurring exclusively in patients with renal disease. Until recently, it has been considered a fibrosing disorder essentially confined to the skin and underlying superficial soft tissue. Recent reports, however, have described patients with involvement of other organ systems, suggesting that this disorder is actually a systemic disease with preferential cutaneous manifestations. We describe a patient with end-stage renal disease with diagnosed NFD/NSF who subsequently developed respiratory failure leading to his death. Autopsy findings showed NFD/NSF involving the skin of all extremities, as well as diffuse diaphragm involvement.

Diaphragm↗
PubMed2005

Idiopathic fibrosing pancreatitis in a 3-year-old girl: a case report and review of the literature.

Idiopathic fibrosing pancreatitis is a rare condition that affects children and adolescents. It can be the cause of recurrent abdominal pain and obstructive jaundice. There are 46 cases reported in the literature, including the first case reported by Comfort et al (Comfort MW, Gambill EE, Baggenstoss AH. Chronic Relapsing Pancreatitis. Gastroenterology 1946;6:239-285) in 1946. We report the case of a 3-year-old girl who presented with abdominal pain and obstructive jaundice. We performed a magnetic resonance cholangiopancreatography study as part of the workup of obstructive jaundice. We will review the literature on pediatric idiopathic fibrosing pancreatitis and highlight the use of endoscopic retrograde choledocho-pancreatography and magnetic resonance cholangiopancreatography in the investigation of this disease.

Abdominal Pain↗
PubMed2006

Idiopathic fibrosing pancreatitis in children--a single-center experience.

PURPOSE: The experience of a single institution on idiopathic fibrosing pancreatitis (IFP) is presented. METHODOLOGY: This is a retrospective review of medical records of affected patients. RESULTS: There were 7 cases with a mean age of 7 years. Upper abdominal pain followed by jaundice was the most common presentation. One child had varicella and 1 developed Crohn's disease 3 years later. In 5 cases, diagnosis was established intraoperatively, whereas 2 cases were diagnosed preoperatively. Ultrasonography suggested the diagnosis in 2 of the 7 cases, contrast computed tomography scan in 1 of the 3 cases, and magnetic resonance cholangiopancreatography in 1 of the 4 cases. Six patients were treated by biliary enteric bypass surgery. Treatment by endoscopic biliary stenting was successful in one. There were no postoperative complications. Pancreatic biopsies showed fibrosis of exocrine elements with preservation of islets. Three patients have pancreatic atrophy, and none has diabetes at follow-up (mean, 62 months). DISCUSSION: Idiopathic fibrosing pancreatitis presents as biliary obstruction in children. Precise preoperative diagnosis of IFP is difficult. Noninvasive imaging has limited sensitivity. Surgery offers satisfactory long-term relief of biliary obstruction. Treatment using temporary endoscopic biliary drainage appears promising in treatment of IFP. Patients should be followed up for pancreatic insufficiency, long-term biliary obstruction, and inflammatory bowel disease.

Adolescent↗
PubMed1994

Observer variation in pattern type and extent of disease in fibrosing alveolitis on thin section computed tomography and chest radiography.

In fibrosing alveolitis the pattern type on thin section computed tomography (CT) predicts histological appearances at open lung biopsy and the likelihood of response to treatment. To test the level of inter- and intra-observer variability on CT and chest radiography (CXR), the pattern type and extent of disease were assessed by four observers (two experienced, two inexperienced). A total of 126 CT examinations and 108 concurrent postero-anterior chest radiographs were scored on two occasions, at least 8 weeks apart. A confidence rating was assigned to each observation. Three out of four observers agreed on pattern type in 81% of cases on CT compared with 54% on CXR (kappa coefficient 0.48 and 0.16 for CT and CXR, respectively). Inter-observer variability in categorizing pattern type on CT was lowest in patients with the highest confidence scores (kappa = 0.63). Confident observations were associated with extensive or moderately extensive disease (P < 0.001), and with a predominantly reticular pattern (P < 0.0001). Intra-observer variability for pattern type on CT was less for the experienced observers (kappa = 0.78 and 0.70) than for the inexperienced group (kappa = 0.50 and 0.37). Inter-observer variability for extent of disease was significantly less on CT than on CXR (standard deviations 7.8% and 9.2% respectively, P < 0.001). This study shows that observer variability using a clinical grading system is lower with CT than with chest radiography in fibrosing alveolitis.

Attitude of Health Personnel↗
PubMed1996

Pulmonary sarcoidosis mimicking cryptogenic fibrosing alveolitis on CT.

AIM: The high resolution CT (HRCT) characteristics of fibrosing alveolitis and sarcoidosis are well established, and HRCT has been shown to have a high diagnostic accuracy in these two conditions. We report two cases of sarcoidosis with features in common with the usual appearances of cryptogenic fibrosing alveolitis (CFA). PATIENTS AND METHODS: Two adult male patients with sarcoidosis established by transbronchial biopsy or open lung biopsy who had undergone HRCT scanning were reviewed. At presentation both cases had an atypical pattern of radiological abnormality more in keeping with the features of CFA. Two observers independently assessed the CT scans for accepted features of CFA and sarcoidosis. RESULTS: In both cases CFA was the most likely radiological diagnosis based on the accepted HRCT criteria for this condition. In neither patient were the appearances suggestive of sarcoidosis. In both cases the predominant distribution of abnormality was peripheral, subpleural and in the lower lung zones rather than peribronchial, central and in the mid and upper lung zones. Nodular opacities were not present in either case. CONCLUSION: HRCT is an accurate and reliable method of suggesting or confirming the diagnosis of CFA and sarcoidosis. In a very small number of cases, sarcoidosis may produce HRCT appearances almost identical to CFA.

Aged↗
PubMed1998

Fibrosing mediastinitis: CT and MR findings.

The aim of this study is to present the computed tomography (CT) and magnetic resonance (MR) findings in three cases of fibrosing mediastinitis, probably secondary to tuberculosis. Two patients had diffuse involvement of the mediastinum and one patient presented with a posterior mediastinal mass. On CT, the patients with diffuse involvement showed infiltrating soft-tissue masses with multiple foci of dense calcifications. One of these lesions enhanced mildly with contrast and one did not enhance. On MR examination, diffuse masses were of heterogeneous, intermediate and low signal on T1-weighted images (n = 2) and were of homogeneous low signal (n = 1) or heterogeneous (n = 1) signal intensity on T2-weighted images. After administration of Gd-DTPA, these mediastinal masses showed heterogeneous enhancement. The posterior mediastinal mass showed homogeneous pronounced enhancement on CT and was homogeneous with intermediate T1 signal intensity and hypointense T2 signal intensity. The CT findings of an infiltrative or well-defined mediastinal mass with scattered calcifications should suggest the diagnosis of fibrosing mediastinitis in the appropriate clinical setting. Although the appearance of calcification on MR are not specific, this technique provides further information than CT especially in those cases in which the lesions appear noncalcified, because MR imaging revealed areas of low signal intensity characteristic of fibrous tissue.

Adult↗
PubMed1998

Mediastinal lymphadenopathy in cryptogenic fibrosing alveolitis: the effect of steroid therapy on the prevalence of nodal enlargement.

PURPOSE: To determine the prevalence of enlarged mediastinal lymph nodes in cryptogenic fibrosing alveolitis (CFA) and to assess whether this frequency was related to steroid administration. MATERIAL AND METHODS: Retrospective review of chest radiographs and high-resolution computed tomography (CT) of 54 patients with proven cryptogenic fibrosing alveolitis (CFA). Twenty-two of the 54 patients received oral steroids up to 2 months before the time of high-resolution CT examination (group 1); the remaining 32 patients had not taken steroids for at least 6 months before study entry (group 2). The prevalence, distribution, and size of enlarged mediastinal lymph nodes was determined in all groups. The diagnosis of CFA was histologically established in 44 patients. In 10 patients the diagnosis of CFA was based on clinical and high-resolution CT findings. RESULTS: Mediastinal lymphadenopathy was present in 26 of 54 patients with CFA. The prevalence of enlarged nodes was 14% (three of 22) in patients who had received oral steroids up to 2 months before the time of CT examination, and 71% (23 of 32) in patients that had not taken steroids for at least 6 months before study entry. CONCLUSION: We conclude that patients with CFA on steroid therapy had a significantly lower prevalence of mediastinal adenopathy (P < 0.001) than patients who had not taken steroids.

Aged↗