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At least 37 records · Page 2Linked to original sources
PubMed1978

Long-term colchicine therapy of familial Mediterranean fever.

Familial Mediterranean fever is a disorder characterized by recurrent fever and polyserositis. Continuous prophylactic colchicine therapy has been effective in suppressing attacks in affected adults. From 30 children with FMF, 14 were selected for colchicine therapy. Eight children continued prophylactic colchicine therapy for 29 months (mean) and experienced a marked decrease in the frequency of attacks. Six other children did not comply with the treatment regimen. Although no deleterious side effects were noted, the safety of long-term colchicine administration in childhood is unknown.

Adolescent↗
PubMed1977

Protracted arthritis in familial Mediterranean fever.

A review of the files of familial Mediterranean fever (FMF) confirmed the rarity of patients suffering protracted arthritic attacks and the propensity of the joints, in general, to recover. While 70% of those afflicted suffered bouts of synovitis, only 57 patients (5% of the FMF-population) experienced protracted attacks involving a total of 84 joints, 36 of them knees and 25 hips. Functional and, usually, anatomical integrity was regained in all but 27 joints. Of the 27 joints producing residual incapacity, 21 were hips. Seven hips showed roentgenologically typical aseptic necrosis of the femoral head and 14 only sclerosis and narrowing of the joint space. Eight hips eventually required total prosthetic replacement. We suggest that the poor prognosis of the hip, in contrast to other joints affected by protracted FMF-arthritis, is related not directly to the metabolic aberrration underlying the disease but to attenuation of the arterial blood supply of the femoral head by synovial exudation. Early aspiration of exudate could alter the prognosis by preventing the complication of aseptic necrosis.

Adolescent↗
PubMedDate not supplied

A cytogenic evaluation of long-term colchicine therapy in the treatment of Familial Mediterranean fever (FMF).

Thirty-eight patients suffering from Famlial Mediterranean Fever (FMF) and undergoing colchicine therapy for periods varying from one week to three years were examined cytogenetically. Preparations were derived from short-term lymphocyte cultures; mitotic rate, percent tetraploidy, and chromosome breakage rates were determined. Twenty-one patients were examined prior to treatment, 22 during treatment and 5 both before and during treatment and 5 both before and during treatment. No statistically significant differences were observed in the parameters studied between ten controls and the patient groups. An in vitro experiment indicated a direct correlation between increased colchicine concentration and mitotic rate. However, tetrapoloidy or chromosome damage showed no such association with colchicine concentration. Among the patient group, pregnancy occurred in four patients while under treatment; three pregnancies resulted in the birth of normal children while the fourth has not yet been completed. In one preganancy, cultured fetal amniotic fluid cells demonstrated no effect of colchicine on the cytogenetic parameters invetigated. These results indicate no untoward effects on long-term colchicine treatment in FMF with respect to fertility, teratogenicity and chromosomal damage.

Adolescent↗
PubMedDate not supplied

Monocyte function in familial Mediterranean fever.

Monocytes derived from the peripheral blood of patients with familial Mediterranean fever (FMF) demonstrated a consistently lower phagocytic capacity (38-44%) for 125I-labelled Shigella flexneri when compared to monocytes from healthy subjects. Phagocytosis of both viable and killed Staphylococcus albus was similar in patients and controls. However, FMF monocytes had a two- to eight-fold depressed bactericidal capacity against S. albus in comparison to normal monocytes. Acid phosphatase and beta-glucuronidase monocyte activities were similar in patients and controls. It is suggested that the defects in monocyte function may be of importance in the pathogenesis of FMF. Colchicine had no effect on any of the indices studied.

Adolescent↗
PubMed1979

Amyloid goitre in familial Mediterranean fever.

Three patients known to suffer from familial Mediterranean fever (FMF), systemic amyloidosis and chronic renal failure developed large amyloid goitres. Amyloid goitre is an extremely rare complication of systemic amyloidosis not previously described in FMF. The clinical and pathological features of these three cases were similar to those previously described in amyloid goitre. In two of the patients abnormalities in thyroid function were consistent with those documented in chronic renal failure. There was evidence of hypothyroidism in a third patient. There was no evidence of amyloid induced dysfunction of other endocrine organs.

Adult↗
PubMed1977

Efficacy of intermittent colchicine therapy in familial Mediterranean fever.

Nine patients with familial Mediterranean fever (FMF) were admitted to a controlled, double-blind trial to determine if there are patients with this disease who are able to abort their acute episodes of pain and fever with short courses of colchicine taken at the onset of attacks. Five patients completed their treatment assignments, and colchicine was significantly effective in aborting the attacks of three but was ineffective in two. The remaining four patients could not be assessed because of insufficient numbers of courses. During the 10 months of the trial, 28 courses of colchicine and 31 of placebo were taken during the early stages of FMF attacks. Twenty-one (75%) colchicine courses were followed by attacks considered to have been aborted, compared to only three (10%) placebo courses. This trial shows that patients can recognize the prodrome of their FMF attacks and that some patients can consistently abort their attacks with short courses of colchicine taken at the very onset of symptoms.

Adolescent↗
PubMed1977

Amyloid deposition in a renal transplant in familial Mediterranean fever.

A patient with familial Mediterranean fever and amyloidosis who received a cadaver renal transplant 6 1/2 years ago was studied to determine the relation of the serum precursor of secondary amyloid (SAA) to the clinical course and to the deposition of amyloid in the transplant. Amyloid fibrils extracted from the patient's kidneys contained protein AA as a major constituent, which identified the amyloid as secondary. Protein AA antiserum was used in an indirect immunofluorescent technique to stain amyloid deposits in sections of the original kidney. A renal biopsy at 2 years showed no amyloid, but a renal biopsy at 4 years showed amyloid. Serum levels of SAA from 3 years before transplant to 6 years after transplant were elevated throughout most of the course.

Adult↗
PubMed1979

Renal transplantation in the amyloidosis of familial Mediterranean fever. Experience in ten cases.

Ten patients with familial Mediterranean fever (FMF) and histologically confirmed amyloidosis received cadaver kidney transplants for treatment of terminal renal disease. Colchicine, 1 mg daily, was included in the routine postoperative regimen from 1974 for amyloidotic patients. Graft and patient survival were compared with ten nonamyloidotic recipients of renal grafts matched for age, sex, type of allograft, and HLA compatibility. In the FMF group, five of ten grafts have survived from 20 to 64 months; in the control group, six of ten. While only recipients with functioning grafts survived in the FMF group, patient survival in the control group is eight of ten after one year. In all five FMF survivors, graft function is satisfactory, proteinuria is absent, and blood creatinine levels are normal. Amyloid involvement of an allograft was documented 16 months after transplantation in the only patient whose maintenance colchicine dosage had been reduced to 0.5 mg daily.

Adult↗
PubMed1978

Unusual immunologic findings in familial Mediterranean fever.

Familial Mediterranean fever (FMF) is an inherited disease of unknown etiology. We report a case in which, during an acute febrile attack, rheumatoid factor and immunoglobulin levels rose, and the levels of complement components fell. The level of urinary fibrinogen degradation products also increased, and all results of tests returned to normal at the end of the acute attack. This suggests that an immunologic phenomenon may play a substantial role in the etiology of FMF.

Adult↗
PubMed1977

Constrictive pericarditis in familial Mediterranean fever.

Fibrosing peritonitis and constrictive pericarditis occurred in an 18-year-old patient who manifested the classic features of familial Mediterranean fever (FMF). Pericardial calcification had been present in chest X-rays taken when the patient was five years old. Intermittent intestinal obstruction and congestive heart failure were relieved by appropriate surgical intervention, but attacks of FMF subsided only after colchicine therapy. This is the first instance of nonuremic pericarditis in our experience with over 1,000 FMF patients. Critical analysis of the reported cases in which pericarditis was attributed to FMF strengthens our belief that the occurrence of pericarditis in a patient with FMF probably represents a fortuitous intercurrent disease.

Adolescent↗
PubMed1977

Renal vein thrombosis as the major cause of renal failure in familial Mediterranean fever.

Eighteen out of 57 patients (31-6 per cent) suffering from Familial Mediterranean Fever (FMF) were found to have the nephrotic syndrome, histologically proven amyloidosis and progressive renal failure. In 14 cases renal function deteriorated rapidly after the first appearance of significant proteinuria, and 12 cases (66-7 per cent) required regular haemodialysis. Seven of these patients, seen in the early stages of renal impairment, were subsequently diagnosed clinically as probably having developed renal vein thrombosis. There was radiological proof of intrarenal or major renal vein occlusion in five which in one patient progressed to inferior vena cave obstruction. Treatment with heparin, plasminogen activators and fibrinogenolytic agents was disappointing although renal function has stabilized in one patient on long term oral anticoagulant therapy. It is suggested that renal vein thrombosis is common in FMF with renal amyloidosis and usually causes rapid deterioration of function and irreversible renal failure requiring dialysis. Renal phlebography may delineate clot in the main renal veins or indicate areas of reduced blood flow due to thromboses in intrarenal venules. Treatment is only partially satisfactory but there is some evidence to suggest that renal phlebography should be undertaken promptly when renal function begins to fall followed by anticoagulant therapy to prevent further thromboembolic complications.

Acute Kidney Injury↗
PubMed1976

Mediterranean lymphomas with alpha heavy chain monoclonal gammopathy.

Nine cases of Mediterranean lymphoma with alpha heavy chain disease are described. All patients were young, aged between 11 and 22 years. Neither sex predominated in the series. The clinical presentation of the disease was either gastrointestinal, with chronic diarrhea and malabsorption, or neoplastic, with tumors frequently associated with either obstructive or perforative complications. Finger clubbing and retardation of physical growth (height and weight) and secondary sexual maturation were usually evident. The laboratory manifestations of the malabsorption syndrome were scant and generally not specific. The intestinal lesions predominated in the beginning of the small bowel, and their gross appearance varied from one case to another: diffuse thickening of the intestinal wall, alone or associated with tumors, or isolated segmentary tumors. Massive mesenteric lymph node enlargement was present in all cases; in some cases hepatic, splenic, or extra-abdominal lymph node spread was noted. Histologically, these lesions demonstrated a varied cellular morphology, but seemed to belong constantly to the B-lymphocytic line. The secretion and excretion by these cells of an abnormal IgA (devoid of light chains), which was revealed by immunoelectrophorectic study of the patients' sera, was the main biochemical abnormality of the disease. This disease presents several problems: 1) its relationship with other intestinal lymphomas; 2) the nature and the degree of malignancy of the intestinal cellular infiltrates; and 3) its identification as a clinicopathologic entity, with the possibility that infectious and genetic factors are significant in its etiology and pathogenesis.

Abdominal Neoplasms↗
PubMed1976

Primary intestinal lymphoma of "Western" and "Mediterranean" type, alpha chain disease and massive plasma cell infiltration: a comparative study of 37 cases.

The clinical and pathological features of 17 "Western" type primary abdominal lymphomas (WTL) are compared with 14 of "Mediterranean" type (MTL). The MTL involved only young adult Mulatto and African patients in whom malabsorption and abdominal pain were the major clinical features. The WTL also predominantly affected Mulatto patients but four cases occurred in Caucasians, and the mean age at presentation was two decades later. An obstructive presentation was the most common; only one patient in this group had evidence of malabsorption. The WTLs were located mainly in the distal small bowel and were nearly all of monomorphic lymphocytic or histiocytic type. The MTLs were sited mainly in the duodenum and jejunum and were of an unusual pleomorphic histologic type. A spectrum of cells from those resembling atypical lymphocytes to large histiocytic types were seen, some of the latter resembling Reed-Sternberg cells. The pattern of mesenteric node infiltration in the MTLs was also unusual in that preservation of the medullary sinuses was a common finding. A notable feature of the MTLs was the presence of a heavy infiltration of mature-looking plasma cells associated with a fairly severe villous atrophy in the lamina propria of the small bowel. In the WTLs the adjacent small bowel did not show this feature. In addition to the above cases three patients with a similar heavy plasma cell infiltrate and villous atrophy but without evidence of a lymphoma are described. These cases may represent examples of MTL in a pre-malignant phase. Also included in this study are three patients with alpha-chain disease (alpha-CD), all with a heavy plasma cell infiltration and villous atrophy of the lamina propria and a pleomorphic type lymphoma involving the mesenteric nodes in all, and the small bowel in two. The lymphomas in alpha-CD have been interpreted as immunoblastic sarcoma by Lukes and Collins. Both genetic and environmental factors may be operative in the MTLs including the cases of alpha-CD.

Adult↗
PubMed1978

"Mediterranean abdominal lymphoma" or immunoproliferative small intestinal disease. Part II: pathological aspects.

The pathology of 25 cases of Mediterranean abdominal lymphoma, better designated as immunoproliferative small intestinal disease (IPSID), are reported from the American University of Beirut Hospital. The series includes nine cases with documented alpha heavy chain disease (alpha-HCD). The disease is characterized by the presence of a diffuse and compact bandlike lymphoplasmacytic infiltration of the proximal small intestinal mucosa. The presence of a concomitant malignant lymphoma in the intestine and/or mesenteric lymph nodes, and of alpha-heavy protein in the serum is commonly encountered. Two histopathologic variants of IPSID are present. The first is characterized by the diffuse infiltration of the mucosa, at sites away from tumoral masses, by either pure plasmacytic infiltration, or mixed lymphoplasmacytic infiltration. This variety is associated with the immunoblastic sarcoma type of malignant lymphoma, and with alpha chain disease (alpha-HCD). The second variant is characterized by a diffuse follicular lymphoid hyperplasia pattern in the small intestinal mucosa. The associated malignant lymphoma is diffuse and undifferentiated often having a starry-sky pattern. This variety is not associated with alpha-HCD. Both histologic variants share the same clinical antecedents. In five patients, mesenteric lymph nodes harbored immunoblastic sarcoma while the intestinal mucosae of the same patients were involved with a benign appearing lymphoplasmacytic infiltration. This finding stresses the need for staging laparatomy. Three patients, with alpha-HCD, had peripheral lymph node involvement with immunoblastic sarcoma. The disease apparently evolves in two stages: an immunoproliferative phase, probably reversible, and a later development of malignant lymphoma. The term immunoproliferative small intestinal disease accurately describes the nature of the entity.

Female↗
PubMed1978

Evidence of acquired immune deficiencies in Mediterranean lymphoma. A possible aetiological link.

Patients with Mediterranean lymphoma (M.L.) had a significant reduction in humoral immunity (IgG and IgM) as well as impaired cellular immunity (50% were anergic to three antigens--P.P.D., mumps, and dinitrochlorobenzene). Any hypothesis for the pathogenesis of M.L. has to account for the peculiar geographic distribution of the disease, the age and sex incidence, the plasma-cell nature of the tumours, the associated heavy plasmacytic proliferation with relatively intact intestinal mucosa, involvement of the proximal small intestine, and alpha-chain production in a large proportion of patients. All areas in which M.L. is common are currently involved in the seventh cholera pandemic. Vibrio cholerae toxin inhibits both immediate and delayed immune reaction in vitro through its effect on cyclic adenosine monophosphate. V. cholerae antigens stimulate the proliferation of IgA-producing immunocytes in the mucosa without deeply penetrating the mucosa. The proximal small bowel is usually affected by the disease but there is little epithelial damage. The population in endemic areas is continuously exposed to V. cholerae antigens and toxins. It is suggested that such exposure, under certain genetic or other circumstances, may produce a state of immunosuppression in the gut thus accelerating, predisposing to, or producing lymphoplasmacytic neoplasia.

Antigens, Bacterial↗
PubMedDate not supplied

Thynnascaris rhacodes sp. n. (Nematoda: Ascaridoidea) in fishes from the Israeli Mediterranean coast.

Thynnascaris rhacodes sp. n. infects Pelates quadrilineatus, Solea vulgaris, Boops boops, Lithognathus mormyrus, Obleida melanura, Diplodus vulgaris, D. sargus, and Sparus auratus from the eastern Mediterranean Sea. Unique in that the cuticle on the female tail is wrinkled, the ascaridoid is further characterized by possessing 27 to 35 precloacal papillae, a body length of 11 to 85 mm, similar spicules 3 to 6% of body length, a length-ratio of intestinal cecum to ventricular appendage of 1 : 1 to 5, and lips longer than wide.

Animals↗