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PubMed2000

Adult height and cryptogenic fibrosing alveolitis: a case-control study using the UK general practice research database.

BACKGROUND: The reasons why cryptogenic fibrosing alveolitis has emerged as a new clinical entity during the second half of the 20th century are unclear. Some environmental exposures have been identified as potential risk factors including occupational dust, cigarette smoking and antidepressants, but there have been no studies of the role of early life exposures. Since adult height reflects, in part, early life experience, we have examined the relation between adult height and the risk of cryptogenic fibrosing alveolitis. METHODS: A case-control study of 569 cases and 3669 age, sex, and community matched controls drawn from the UK General Practice Research Database was undertaken. RESULTS: Evidence was found of an inverse association between quintile of height and cryptogenic fibrosing alveolitis (odds ratio (OR) per increase in height quintile 0.93, 95% CI 0.86 to 0.99). This association was not diminished by adjustment for smoking status (OR 0.93, 95% CI 0.87 to 1.00), but some minor attenuation did occur after adjustment for oral corticosteroid use (OR 0.94, 95% CI 0.88 to 1.02). There was a significant interaction with sex such that the effect of height was strong in women (OR 0.85, 95% CI 0.75 to 0.97) and absent in men (OR 1.00, 95% CI 0.91 to 1.09). CONCLUSIONS: These findings raise the possibility that early life exposures may be important in determining the lifetime risk of developing cryptogenic fibrosing alveolitis.

Aged↗
PubMed1983

The significance of antinuclear and DNA antibodies in cryptogenic fibrosing alveolitis.

The roles of antinuclear and DNA antibodies in the pathogenesis of cryptogenic fibrosing alveolitis were investigated in 53 patients. Twenty-two patients who had antinuclear antibodies detected in their serum had a significantly higher proportion of women, a higher prevalence of Raynaud phenomenon and digital vasculitis, and higher erythrocyte sedimentation rates, paralleled by raised serum globulin and IgG levels, than patients with no antinuclear antibodies detected. Serum antibodies to double-strand DNA (DS-DNA), assayed by a Farr binding technique, were significantly raised in 25% of patients with cryptogenic fibrosing alveolitis. Serum binding of single-strand DNA (SS-DNA) was greatly increased in all the patients with cryptogenic fibrosing alveolitis, achieving levels similar to those found in systemic lupus erythematosus. Serum DS-DNA binding correlated with IgA levels but not with disease activity. Thus, unlike in systemic lupus erythematosus, antibodies to DS-DNA and SS-DNA with their capacity to form immune complexes are unlikely to be of major importance in the pathogenesis of cryptogenic fibrosing alveolitis.

Adolescent↗
PubMed1993

Aerosolised ribavirin in patients with advanced cryptogenic fibrosing alveolitis: a pilot study.

BACKGROUND: A report has recently been published concerning a patient with a cryptogenic fibrosing alveolitis who showed a striking improvement after being treated with the antiviral drug ribavirin (tribavirin, Virazid). The objective of this study was to further evaluate, in an open trial, the efficacy of rivabirin in cryptogenic fibrosing alveolitis. METHODS: Ten patients (eight women) with advanced cryptogenic fibrosing alveolitis received aerosolised ribavirin (6 g/day for 15 days). Chest radiographs, lung function, and severity of dyspnoea were evaluated before and after two weeks of rivabirin treatment and also at three and 12 months. RESULTS: No differences in radiographs, lung function impairment, or severity of dyspnoea were found after treatment. No side effects were detected. CONCLUSIONS: Administration of high doses of aerosolised ribavirin has no beneficial effects in patients with advanced cryptogenic fibrosing alveolitis.

Aerosols↗
PubMed1993

Fibroblast chemotactic response elicited by native bronchoalveolar lavage fluid from patients with fibrosing alveolitis.

BACKGROUND: In fibrosing alveolitis activation of lung fibroblasts is the decisive event in the pathogenetic sequence leading to pulmonary fibrosis. Fibroblast stimulating activity was measured in bronchoalveolar lavage (BAL) fluid to assess its relationship to the activity of fibrosing alveolitis. METHODS: Nine control subjects and 40 patients with fibrosing alveolitis caused by idiopathic pulmonary fibrosis (n = 22) or pulmonary involvement in systemic sclerosis (n = 18) were studied. All patients were followed up by lung function testing for a minimum of six months (mean (SE) 13.3 (1.4) months). Twenty five patients received immunosuppressive therapy and 15 refused. At the beginning of follow up BAL was performed and, as a possible indicator of fibroblast stimulating mediators within the lungs, chemotactic migration of cultured human fibroblasts elicited by native BAL fluid was measured in Boyden-type chambers and expressed as a percentage of the chemoattractant effect of 25 ng/ml platelet derived growth factor. The procollagen III peptide level in BAL fluid served as a marker for collagen synthesis. RESULTS: Chemoattractant activity was elevated in the patients with idiopathic pulmonary fibrosis and systemic sclerosis compared with the control group, (mean (SE) 56.4% (8.5%)) and 72.3% (16.3%) v 12.6% (4.0%). Chemoattractant activity was inversely correlated with total lung capacity (TLC) (r = -0.45) and with vital capacity (VC) (r = -0.33). Procollagen III peptide concentrations in BAL fluid and chemoattractant activity were not significantly correlated. For further evaluation chemoattractant activity of 36% (mean value of controls +2 SD) was used to separate normal (< 36%) from elevated (> or = 36%) activity. At the end of follow up, untreated patients with high chemoattractant activity (> or = 36%) showed a significant reduction of VC, TLC, and exercise arterial oxygen tension (PaO2) and a small decrease in carbon monoxide transfer factor (TLCO), whereas a significant improvement in VC, TLC, and TLCO and a small increase of exercise PaO2 occurred in treated patients with high chemoattractant activity. Patients with low chemoattractant activity (< 36%) showed no consistent change in lung function measurements, irrespective of treatment. In contrast, lung function results and differential cell counts in BAL fluid failed to identify progressive disease. CONCLUSIONS: In patients with fibrosing alveolitis the chemoattractant activity of BAL fluid seems to be an independent indicator of lung fibroblast stimulating activity providing relevant information about disease activity, and may help to improve the clinical management of these patients.

Adult↗
PubMed2004

Aneurysmal bone cysts in children: complications of fibrosing agent injection.

PURPOSE: To report complications of direct fibrosing agent injection in the treatment of aneurysmal bone cysts (ABCs) in children. MATERIALS AND METHODS: The authors retrospectively analyzed all cases of ABCs treated with direct fibrosing agent injection (Ethibloc; Ethnor Laboratories, Ethicon, Noderstedt, Germany) at Robert Debré Hospital since 1994. Histologic diagnosis was assigned by means of surgical biopsy findings prior to treatment. Treatment responses were categorized. Injection was administered with general anesthesia, computed tomographic guidance, and use of a 14- to 16-gauge needle. Contrast material was injected to determine presence of intracystic septa and verify absence of venous opacification. Amount of fibrosing agent injected corresponded to amount of contrast material necessary to fully opacify the cyst. Intraosseous needle track was obliterated with histoacryl injection. RESULTS: Fifteen patients were treated. Mean follow-up was 80 months; no patient was lost to follow-up. One patient experienced pulmonary embolus that necessitated a 7-day intensive care unit stay. Four patients experienced early aseptic fistulization after the first injection, which led to surgical débridement and curettage. Five patients had transient inflammatory reaction with mild 38 degrees C fever, which was controlled with analgesic and antiinflammatory drugs. Eleven patients did not require surgery, and results at latest follow-up were considered to indicate complete healing (type 1 results) in nine and incomplete healing (type 2 results) in two. For type 1 results: Six patients received one injection, two received two injections, and one received three injections. For type 2 results: one patient received one injection, and one received three injections. CONCLUSION: A high rate of major local and general complications was encountered with use of direct fibrosing agent injection; the technique has been abandoned for treatment of ABCs.

Adolescent↗
PubMed1995

Increased oxidation of extracellular glutathione by bronchoalveolar inflammatory cells in diffuse fibrosing alveolitis.

An unbalanced oxidative stress is thought to be an important element in the pathogenesis of diffuse fibrosing alveolitis (DFA). The purpose of our study was to investigate the role of reactive oxygen metabolites (ROMs) released from cultured bronchoalveolar inflammatory cells (BA-cells) on glutathione oxidation. We studied bronchoalveolar lavage samples from 10 healthy controls and from 20 patients with diffuse fibrosing alveolitis (all were nonsmokers). BA-cells obtained by bronchoalveolar lavage (BAL) were incubated with 50 microM of reduced glutathione (GSH). Oxidation of GSH to glutathione disulphide (GSSG) by BA-cell derived oxidants was detected as a decline of GSH in the supernatants. Total glutathione (GSHtot = GSH + 2 GSSG) and GSSG in the epithelial lining fluid (ELF), and methionine sulphoxide (Met(O)) content of BAL proteins were determined. In diffuse fibrosing alveolitis the oxidative activity of BA-cells was enhanced, GSHtot and GSH were decreased, whereas the GSSG:GSH ratio was increased. The oxidative activity of BA-cells correlated positively with the GSSG:GSH ratio, but not with the methionine sulphoxide content. The methionine sulphoxide content was elevated in diffuse fibrosing alveolitis and inversely correlated with GSHtot. The methionine sulphoxide content also correlated positively with the percentage of BAL neutrophils. We conclude that BA-cell-derived reactive oxygen species are capable of oxidizing extracellular GSH in vitro. The positive correlation between the BA-cell oxidative activity in vitro and GSSG:GSH ratio in ELF suggests that a similar oxidative effect on extracellular GSH may also occur in vivo.(ABSTRACT TRUNCATED AT 250 WORDS)

Adult↗
PubMed1997

Functional impairment in fibrosing alveolitis: relationship to reversible disease on thin section computed tomography.

Thin-section computed tomography (CT) provides a reproducible method of quantifying global disease extent and can also discriminate between fibrotic disease, with predominance of reticular abnormalities, and reversible inflammatory cell infiltration, shown as ground-glass attenuation. The aim of this study was to determine whether functional impairment varied according to the presence of ground-glass attenuation on CT, independently of extent of disease on CT, demographic factors, smoking history, therapeutic status, and the type of fibrosing alveolitis (lone cryptogenic fibrosing alveolitis (CFA) versus fibrosing alveolitis associated with systemic sclerosis (FASSc)). Patients with concurrent emphysema on CT (n = 16) and FASSc patients with end-stage pulmonary hypertension (n = 5) were excluded. One hundred and eleven patients were studied (CFA, n = 54; FASSc, n = 57). The severity of functional impairment did not vary independently with the presence of predominant ground-glass attenuation, mixed appearance and predominant reticulation on CT. In 34 treated patients undergoing serial CT scanning, the severity of functional impairment did not differ independently between patients with and without regression of ground-glass attenuation at follow-up. We conclude that the severity of functional impairment does not discriminate between inflammatory and fibrotic disease in fibrosing alveolitis, as judged by initial and serial computed tomographic scanning, after adjustment for the morphological extent of disease on computed tomography.

Female↗
PubMed1998

Survival in patients with cryptogenic fibrosing alveolitis: a population-based cohort study.

STUDY OBJECTIVES: To determine the median survival of patients with cryptogenic fibrosing alveolitis, in comparison to that expected of individuals of the same age and sex from the general population. To estimate the effect of survival bias incurred by studying both incident and prevalent cases. To identify factors associated with survival. DESIGN: Cohort study. SETTING: Nine hospitals from the Trent Region of England. PATIENTS: Two hundred forty-four cases of cryptogenic fibrosing alveolitis: 168 patients were alive on day 1 of the study (prevalent cases), and 76 patients had newly diagnosed conditions over a prospective 18-month period of patient recruitment (incident cases). MEASUREMENTS: Age, sex, date of diagnosis, lung function at presentation, and details of treatment prescribed were extracted from hospital clinical records. Data on lifetime smoking habits were collected by postal questionnaire. Vital status was established from the general practitioner. The expected survival of each case was extracted from the English life tables. RESULTS: Median survival for incident cases was 2.9 years and for prevalent cases 9 years, compared to expected values of 10 and 13 years, respectively. Incident cases had significantly worse survival than prevalent cases, even after adjustment for the effects of age, sex, smoking history, lung function at presentation, and treatment (hazard ratio, 4.53 [95% confidence interval, 2.71 to 7.56]; p<0.001). Lower FVC at presentation and the use of corticosteroid treatment were both associated with worse survival. CONCLUSIONS: The inclusion of prevalent cases leads to significant overestimation of the median survival for patients with cryptogenic fibrosing alveolitis. In newly diagnosed cases of cryptogenic fibrosing alveolitis, median survival is only 2.9 years, and expected life span is reduced by approximately 7 years.

Adrenal Cortex Hormones↗
PubMed1993

Serial CT in fibrosing alveolitis: prognostic significance of the initial pattern.

OBJECTIVE: In patients with fibrosing alveolitis, a reticular pattern on CT scans of the lungs correlates with histologic findings of fibrosis, whereas a ground-glass pattern has been reported to correlate with reversible inflammatory disease. The majority of patients with fibrosing alveolitis have a ground-glass component visible on CT scans, but response to therapy is infrequent. The aim of this study was to evaluate the prognostic significance of the relative extents of ground-glass and reticular patterns by analyzing serial changes in these CT appearances. SUBJECTS AND METHODS: Serial CT scans were analyzed in 56 patients (21 with idiopathic pulmonary fibrosis and 35 with fibrosing alveolitis associated with systemic sclerosis). The relative extents of ground-glass and reticular patterns were assessed on the initial CT scan. Overall extent of abnormal lung and distribution of disease on initial CT scans were also categorized. Changes in extent and appearance of disease were evaluated in paired anatomically comparable CT sections and assessed independently by two observers; the median interval between scans was 16 months. Serial changes on CT were examined in relation to the initial pattern, extent, and distribution of disease seen on CT scans and in relation to trends in results of concurrent pulmonary function tests. RESULTS: Changes in the extent of disease were due to regression of a ground-glass pattern in 18 patients, an increase in a reticular pattern in nine patients, and an increase in a ground-glass pattern in five patients. A reticular pattern did not regress in any patient. In treated patients, diminution in extent of disease, shown as regression of a ground-glass pattern, was seen most frequently when a ground-glass pattern was the most extensive abnormality at initial scanning (p < .002), independent of extent or distribution of disease seen on CT scans and the type of fibrosing alveolitis. When a ground-glass pattern was associated with an equally extensive reticular pattern, the extent of disease diminished with therapy in a minority of patients (5/13). Improvement in results of pulmonary function tests was associated with regression of a ground-glass pattern in the majority of patients (8/11). CONCLUSION: These findings indicate that the prognostic significance of a ground-glass pattern depends on the extent of an associated reticular pattern and is independent of the extent and distribution of disease.

Female↗
PubMed1987

The fibrositis syndrome: diagnosis, differential diagnosis and pathogenesis.

The fibrositis syndrome represents a clinically definable entity, which is characterised by spontaneous pain, especially in the lumbar and cervical region, and more rarely in other movable parts, multiple tendomyosis, tendinitis and insertion tendinitis, as well as a wide variety of functional syndromes, vegetative symptoms and psychological disturbances. The course of the illness is rather variable; its commencement can be slow to develop or acute, initially presenting a localised clinical picture similar to a lumbar or cervical condition, and gradually, or in jumps, showing a deterioration accompanied by a generalisation of complaints. Various factors are probably instrumental in triggering off the fibrositis syndrome. The most important ones are emotional stress whereby symptoms of fear, depression, etc. lead to muscle tension and insertion tendinitis. Somatic factors such as malposition of the spinal column, may also contribute towards the manifestation of the clinical picture. The so-called secondary fibrositis syndromes should be defined from the point of view of differential diagnosis; they can develop within the framework of inflammatory rheumatic conditions, through infections and endocrinopathy. Differential diagnosis is very difficult considering depression alongside pain in the movable parts and "psychogenic rheumatism". Smooth transitions are in existence. Polymyalgia rheumatica and polymyositis, which produce similar clinical pictures, must be differentiated from the fibrositis syndrome.

Diagnosis, Differential↗
PubMed2006

Multiorgan involvement in nephrogenic fibrosing dermopathy: an autopsy case and review of the literature.

Nephrogenic fibrosing dermopathy is a recently recognized, scleromyxedema-like fibrosing skin condition that occurs in individuals with acute or chronic renal failure. Although the early descriptions of this disorder describe a purely cutaneous disease process, 2 recent autopsy reports have identified apparent multiorgan fibrosis with involvement of skeletal muscle, myocardium, lungs, kidneys, and testes. We describe a 23-year-old man with nephrogenic fibrosing dermopathy and significant fibrosis of the atrial myocardium and dura mater, which was identified at autopsy. Dural fibrosis is a previously undescribed systemic manifestation of nephrogenic fibrosing dermopathy. The literature is reviewed.

Adult↗
PubMed1991

Hepatic histological findings after transplantation for chronic hepatitis B virus infection, including a unique pattern of fibrosing cholestatic hepatitis.

Long-term follow-up of 27 patients with hepatitis B virus-related chronic liver disease treated by transplantation showed that 23 had hepatitis B virus recurrence. In 13 patients late changes in the grafts were similar to those described in other series: minor abnormalities in five cases, chronic active hepatitis in five cases and non-hepatitis B virus-related graft dysfunction in three cases. Three patients had incomplete histological follow-up. Analysis of the histological changes and viral antigen expression in six cases revealed a distinct and novel pattern termed fibrosing cholestatic hepatitis. Development of fibrosing cholestatic hepatitis was associated with rapidly progressive graft dysfunction. It is postulated that this pattern of fibrosing cholestatic hepatitis develops because of a high cytoplasmic expression of viral antigens, including HBsAg. The remaining case had some features of fibrosing cholestatic hepatitis. The main histological features of this unique syndrome include thin, perisinusoidal bands of fibrosis extending from portal tracts to surround plates of ductular-type epithelium; prominent cholestasis; ground-glass transformation; and ballooning of hepatocytes with cell loss and mild mixed inflammatory reaction.

Adult↗
PubMed1989

Medicolegal aspects of fibrositis syndrome.

The recent Australian epidemic of localized fibrositis syndrome is used to highlight the role of compensation and medicolegal systems on the clinical features and outcome of fibrositis syndrome. It is suggested that psychological factors are of great importance when this syndrome occurs after minor injury or de novo in the workplace. Current compensation and medicolegal systems usually negate appropriate management plans and thus lead to perseveration of fibrositis syndrome. It is suggested that public health policies in regard to fibrositis syndrome in this context need reappraisal.

Australia↗
PubMed1988

Fibrositis and primary hypothyroidism.

The prevalence of fibrositis was determined in 100 patients with subclinical or biochemical primary hypothyroidism. Nineteen patients reported symptoms of joint and/or muscle pain with stiffness. Five of these patients presented 7 or more tender points on examination, thus allowing a diagnosis of fibrositis to be made in only 5% of the total group. Symptomatic improvement after thyroid hormone replacement occurred in 10 of the 19 patients, including 3 of those with fibrositis. There were no significant changes in tender points. Our data indicate that fibrositis is uncommon in patients with primary hypothyroidism despite the frequent occurrence of symptoms suggestive of this syndrome.

Adolescent↗
PubMed1988

Sleep and symptoms in fibrositis syndrome after a febrile illness.

Sleep physiology and symptoms of 9 patients with fibrositis syndrome secondary to a febrile illness were compared to 9 patients with fibrositis syndrome who did not attribute their symptoms to a febrile illness and to 10 healthy controls. Both patient groups showed an alpha EEG (7.5 to 11 Hz) nonrapid eye movement sleep anomaly, had similar observed tender points, and self-ratings of musculoskeletal pain. These findings suggest that patients with postfebrile fibrositis have a nonrestorative sleep disorder characteristic of patients with fibrositis syndrome and share similar symptoms with patients who have a "chronic fatigue syndrome."

Adult↗
PubMedDate not supplied

[Relation between pain experience and psychological markers in patients with fibrositis syndrome and patients with rheumatoid arthritis].

We attempted to ascertain to what degree pain experience in two groups of rheumatic patients is influenced by personality traits and psychosocial characteristics. 20 patients with fibrositis syndrome and 20 patients with rheumatoid arthritis completed the Freiburger Personality Inventory, the Giessen Test and the Revised Multidimensional Pain Scale. There was no difference in mean scores of the three questionnaires between the groups but patients with fibrositis syndrome tend to characterize themselves as being psycho-somatically disturbed. The number of significant correlations between pain scales and the personality scales is distinctly smaller in the fibrositis group than in the arthritis group. This result is interpreted as an indication for alexithymia in the fibrositis group.

Arthritis, Rheumatoid↗
PubMed1987

Isometric and isokinetic muscle strength in patients with fibrositis syndrome. New characteristics for a difficult definable category of patients.

A common complaint among patients with fibrositis syndrome is exhaustion and fatique. It was therefore felt desirable to evaluate the muscle strength of these patients compared with normal subjects. Maximum isometric and isokinetic strength of knee extension was measured in 15 patients and 15 healthy matched subjects, using an isokinetic dynamometer (Cybex II). Maximum isometric strength at various knee extension angles (90 degrees, 60 degrees and 30 degrees degrees) was significantly (p less than 0.001) lower in the fibrositis group than in controls, a reduction of approximately 58-66%. Maximum isokinetic strength at various knee extension velocities (30-240 degrees per second) was also significantly (p less than 0.01) lower in the fibrositis group than in controls, the reduction being approximately 41-51%. In conclusion, isometric and isokinetic muscle strength is found to be lower in patients with fibrositis syndrome than in healthy subjects.

Female↗
PubMed1987

Undetected fibrositis in primary care practice.

The prevalence of symptoms related to fibrositis was investigated for patients seen in a primary care setting. Of 692 adult patients evaluated, 33 (4.6 percent) had symptoms of unexplained, chronic, diffuse muscular pain. Only three patients had been diagnosed as having fibrositis by their physician. Eighteen of 31 patients had symptoms sufficiently severe to interfere with their ability to perform their job or household chores. The percentages of these patients who met the fibrositis criteria ranged from 17 to 55 percent. These results suggest that unexplained, diffuse muscular aching is a common problem, that it is rarely diagnosed, and that the use of several criteria to define fibrositis excludes many patients with the typical primary symptoms.

Adult↗