Cystic Bone Lesions Research Articles

Aneurysmal Bone Cyst: the Role of Fibular Graft in its Treatment

Aneurysmal bone cyst is a relatively uncommon benign bone lesion for which curettage and cancellous bone grafting is a widely accepted method of treatment. This paper presents a report of eight patients with aneurysmal bone cysts; four in the humerus, three in the femur and one in the proximal phalanx of the index finger. Seven patients were treated by curettage and cancellous bone graft and one by autogenous fibular graft combined with cancellous allograft. Patients treated by cancellous bone grafting developed recurrence within a year. Three of these recurrent cysts were then treated by autogenous fibular graft combined with allograft. All four patients treated by fibular graft showed satisfactory healing of the cysts with no recurrence at three years' follow-up. Analysis of these patients suggests that recurrence of the aneurysmal bone cyst is very frequent after curettage and cancellous bone grafting. It is recommended that where total excision of the cyst is not feasible, the treatment of choice should be curettage and autogenous fibular bone grafting combined with cancellous bone graft. Removal of the fibula form its middle third does not cause any disability and the fibula regenerates completely within a period of three months.

顎骨に発生した脈りゅう性骨嚢胞の２例

Aneurysmal bone cyst is a benign cystic lesion of bone, and the incidence of this disease in the jaw is low. We reported two cases of aneurysmal bone cyst of the jaw. A 31-year-old woman presented with complaints of pain and swelling at the left side of the face. Since X-ray and CT-scan suggested a cystic lesion, cystectomy was performed. The lesion was histologically diagnosed to be an aneurysmal bone cyst. The other patient, a 40-year-old man, showed swelling at the right mandible. The patient's occlusion was impaired by the gingival swelling. X-ray findings showed the presence of a honeycomb-like lesion at the body and ramus of the mandible. CT-scan findings showed buccal and lingual expansion at the molar region. Microscopic features of the biopsy specimen were consistent with diagnosis of an aneurysmal bone cyst. We performed conservative curettage of the lesion. The postoperative courses of both of these cases were uneventful, and there has been no sign of recurrence after 7 years and 4 years, respectively.

透析肩に対する手術的療法の経験

Long-term hemodialysis related arthropathies in 10 shoulders of 8 patients were treated surgically. The pathological foci were almost all in the intertubercular sulcus where the long tendon brachii inserts and the rotator cuff attaches to the greater tuberosity of the humerus. Marked proliferation of bursae and multiple cystic bone lesions were found with amyloid deposition observed histologically in all cases.Surgical treatment such as curettage and bone grafting were quite effective and the clinical symptoms decreased dramatically after surgery. The enthesopathy induced by amyloid deposition is considered to be the main etiological pathology of the hemodialysis related arthropathy in the shoulder.

Association between Shoulder Pain and Cystic Bone Lesion in Hemodialysis Patients

Association between Shoulder Pain and Cystic Bone Lesion in Hemodialysis Patients

Allograft in the treatment of benign cystic lesions of bone.

Seventeen patients with benign cystic osseous lesions were treated by curettage and grafting using allogenic decalcified bone. Human bones were partially decalcified using 0.6 N HCl and preserved in 90% ethanol in a deep freezer at -16 degrees C. The cystic lesions were: 5 cases of fibrous dysplasia, 4 aneurysmal bone cysts, 3 simple bone cysts, 2 giant-cell tumours, 1 chondromyxoid fibroma, 1 non-ossifying fibroma and 1 fibrous cortical defect. The bones involved were: femur, tibia, humerus, fibula and calcaneum. Infection was a complication in three patients. In two of these it did not interfere with healing, but in one it persisted for more than 1 year with partial resorption of the graft. The time to adequate incorporation of the graft varied from 6 to 9 months in children and 9 to 15 months in adults. The overall response compares favourably with that to allograft from more sophisticated bone banks.

Allogenic decalbone in the repair of benign cystic lesions of bone.

Forty-six cases of benign cystic lesions of bone were treated by curettage and compact filling using partially decalcified allogenic bone graft (Decalbone); of these, 35 were available for study. Decalbone was prepared by partial decalcification with 0.6 N hydrochloric acid (HCl) of human bones generally obtained from freshly amputated limbs. The commonest lesions of bone were giant cell tumours (14) and aneurysmal bone cysts (15), and the commonest bones involved were the femur (23) and the tibia (12). There was one failure and four recurrences. Five cases were infected but this did not interfere with healing of the primary lesion. Radiological incorporation of the graft was seen at about 3 months in unicameral bone cysts, at 4-6 months in aneurysmal bone cysts and at 6-9 months in giant cell tumours. There was no recurrence in any case of giant cell tumour, but three aneurysmal bone cysts recurred. There was no clinical immune reaction.

Generalized amyloidosis from β2-microglobulin, with caecal perforation after long-term haemodialysis

A 73-year-old man with chronic renal failure of undetermined aetiology had received haemodialysis for 12 years when he died of acute purulent peritonitis due to caecal perforation. Amyloid deposits detected in a cystic bone lesion in the left hip had caused a pathological fracture 17 days before death. At autopsy, extensive amyloid deposits were found in the osteoarticular system, in the cartilaginous surface and the capsular tissue of joints, ligaments, vertebral discs and bone. In addition, vascular amyloid deposits were diagnosed in the heart, kidneys, testes, lungs, skin and in the gastrointestinal tract. A special feature of this case were interstitial amyloid deposits forming a fine-meshed structure in the myocardium and plate-like deposits in the gastrointestinal tract. Immunohistochemically, all these deposits reacted strongly with antibody to human beta 2-microglobulin but showed no reaction with antibodies to AA, A-lambda, A-kappa and AF. The present case demonstrates that extra-osteoarticular manifestations of AB-amyloidosis can cause serious complications.

Effect of dialysis membrane and patient's age on signs of dialysis-related amyloidosis

This 12 center study was designed to assess factors affecting the development and progression of beta 2-microglobulin amyloidosis in long-term dialysis. A total of 221 patients who were on hemodialysis for more than five years, and who were treated the entire time only with AN69, a biocompatible, highly permeable membrane, or cuprophane, a less permeable, poorly biocompatible membrane (Cell) were evaluated for time on dialysis, development of carpal tunnel syndrome, and cystic bone lesions. X-ray documentation was taken in a minimum of four of the six following joints: both hips, wrists and shoulders. The data demonstrate that patients treated solely by AN69 membranes display signs of bone amyloidosis less frequently than do those treated by Cell membranes. Age at onset of dialysis was found to have a striking correlation with the development of carpal tunnel syndrome and bone amyloidosis, while no significant influence was found for hyperparathyroidism, sex or year of first dialysis.

Atypical Features of Primary Hyperoxaluria in End-Stage Renal Disease

Two case histories of patients with end-stage renal disease subsequently found to have primary hyperoxaluria are reported. In the setting of renal failure, the diagnosis is both difficult, due to diminished oxalate excretion, and important, because of frequent graft loss due to oxalate deposition after renal transplantation. The diagnosis was obtained by renal and bone biopsies. Plasma oxalate levels were normal in one patient and the other patient presented with extensive cystic bone lesions. Primary hyperoxaluria should be considered whenever nephrocalcinosis and/or nephrolithiasis are associated with end-stage renal disease.

Percutaneous intralesional brushing of cystic lesions of bone: a technical improvement of diagnostic cytology

A modified bronchial brush is presented as a new cytologic method to improve the value of fine-needle aspiration biopsy in the diagnosis of cystic lesions of bone. Four female and six male patients ranging in age from 10 to 44 years were studied. In all cases intralesional brushing was percutaneously performed under fluoroscopic control with local anesthesia. Fine-needle aspiration biopsy preceded intralesional brushing and was performed as described elsewhere. The brush was driven into the walls and septa of an osteolytic lesion through a 14-G needle; material obtained was fixed and processed for cytological and/or histological examinations.

A subset of systemic lupus erythematosus with progressive cystic bone lesions.

Clinical and serological findings of 16 patients with systemic lupus erythematosus (SLE) who had progressive cystic bone lesions were compared with a control group of 19 patients with SLE without radiological evidence of bone cysts. Central nervous system manifestations, synovitis, and other radiologically observed skeletal abnormalities were more prevalent in the patients with cysts than in the control group. Higher concentrations of C reactive protein, and a greater incidence of rheumatoid factor positivity were seen in the patients with cysts than in the control patients, but no other serological differences were found. It is suggested that patients with SLE with progressive cystic lesions form a subgroup of the syndrome characterised by an increased acute phase reaction.

β 2-microglobulin amyloidosis

A number of rheumatic disorders occur in patients on long-term hemodialysis treatment. In recent years a clinical syndrome comprising carpal tunnel syndrome, destructive arthropathy, and cystic bone lesions has been recognized in these patients. Congo-red staining and microscopy in polarized light reveal a high frequency of amyloid in the affected tissues. Amino acid sequence data of the isolated major amyloid fibril protein show its identity with beta 2-microglobulin. beta 2-microglobulin amyloid has a predilection for synovial tissues and bone, but visceral deposits may also occur indicating the systemic nature of the disease. The clinicopathological features, pathogenesis, and diagnosis of beta 2-microglobulin amyloidosis are reviewed, and the therapeutic and prophylactic measures discussed. The identification of beta 2-microglobulin as an amyloidogenic protein has in an important way contributed to the understanding of the mechanisms of amyloidogenesis in general and emphasized the complexity of amyloid disease and the diversity of proteins capable of forming congophilic fibrillar deposits in human tissues.

Inflammation as the Possible Cause of Cystic Radiolucencies in Carpal Bones of Patients on Hemodialysis

In this investigation of the etiology of amyloid-associated arthropathy in hemodialysis (HD) patients, determination of whether amyloid deposition triggered the formation of the cystic radiolucent bone lesions was attempted. The relationship between these lesions in carpal bones and carpal tunnel syndrome (CTS) was studied by radiography and tomography, and one of the carpal bones, the os capitatum, was examined histologically at autopsy. The mean number of cystic radiolucent lesions in carpal bones of the 21 patients with CTS was significantly greater than that in the 43 patients without CTS. On tomograms, cystic radiolucent lesions of carpal bones had interconnections with joint spaces. Inflammatory fibrous granulation tissue, with or without amyloid deposits, was found in the cystic lesions. It was concluded that cystic radiolucent lesions in carpal bones of patients on HD was related to CTS, and that synovitis and subsequent chronic inflammation were responsible. Thus, amyloid deposits did not trigger the formation of such lesions.

Inflammation as the possible cause of cystic radiolucencies in carpal bones of patients on hemodialysis.

In this investigation of the etiology of amyloid-associated arthropathy in hemodialysis (HD) patients, determination of whether amyloid deposition triggered the formation of the cystic radiolucent bone lesions was attempted. The relationship between these lesions in carpal bones and carpal tunnel syndrome (CTS) was studied by radiography and tomography, and one of the carpal bones, the os capitatum, was examined histologically at autopsy. The mean number of cystic radiolucent lesions in carpal bones of the 21 patients with CTS was significantly greater than that in the 43 patients without CTS. On tomograms, cystic radiolucent lesions of carpal bones had interconnections with joint spaces. Inflammatory fibrous granulation tissue, with or without amyloid deposits, was found in the cystic lesions. It was concluded that cystic radiolucent lesions in carpal bones of patients on HD was related to CTS, and that synovitis and subsequent chronic inflammation were responsible. Thus, amyloid deposits did not trigger the formation of such lesions.

Bone cysts and haemodialysis-related amyloidosis

Carpal tunnel syndrome and arthropathy are frequent complications of long-term haemodialysis. Both are associated with cystic lesions of bone and are related to synovioarticular deposition of a new type of amyloid derived from circulating beta 2-microglobulin. Retrospective analysis of the radiographic skeletal surveys of all 58 patients on haemodialysis for 6 or more years revealed one or more cystic bone lesions in 36% of patients. Radiographic analysis indicated that a number of these cysts were due to hyperparathyroid bone disease or joint degeneration, but a majority showed features suggesting an arthropathy of another aetiology. We present evidence that the development of these latter cysts in an individual on long-term dialysis is an indicator of the presence of synovioarticular amyloidosis of the beta 2-microglobulin type. The radiographic characteristics and development of these cysts, which we believe are due to bony infiltration from synovioarticular amyloid are described.

Das intraossäre Ganglion des Tibiakopfes

Intra-osseous ganglia are described relatively rarely. The aetiology and pathogenesis, the incidence, age and sex distribution, clinical and radiological findings, therapy, prognosis as well as the differential diagnosis of this clinical picture are reported with reference to the pertinent literature and a case of this cystic bone lesion in the head of the tibia.

Lack of Amyloid in Abdominal Fat Aspirates From Patients Undergoing Long-term Hemodialysis

Recent reports describe the carpal tunnel syndrome (CTS) due to amyloid infiltration of the beta 2 microglobulin protein as a frequent complication of long-term hemodialysis. Carpal synovial and cystic bone lesion amyloid deposits have been reported; however, the extent of systemic amyloid deposition has not been determined. We examined 30 patients undergoing long-term hemodialysis for CTS and performed abdominal fat tissue aspiration for amyloid staining to evaluate the presence of systemic amyloid disease. In this group, CTS was frequent (37%) and its prevalence correlated with the duration of hemodialysis. In all patients, the abdominal fat tissue, stained with Congo red, was negative for amyloid deposits. These results confirm that CTS is a frequent complication of long-term hemodialysis; however, in this study, no detectable amyloid deposits were found in abdominal subcutaneous fat tissue. Thus, abdominal fat aspiration may not be a reliable screening test for hemodialysis-associated amyloidosis.

Lack of amyloid in abdominal fat aspirates from patients undergoing long-term hemodialysis

• Recent reports describe the carpal tunnel syndrome (CTS) due to amyloid infiltration of the β₂microglobulin protein as a frequent complication of long-term hemodialysis. Carpal synovial and cystic bone lesion amyloid deposits have been reported; however, the extent of systemic amyloid deposition has not been determined. We examined 30 patients undergoing long-term hemodialysis for CTS and performed abdominal fat tissue aspiration for amyloid staining to evaluate the presence of systemic amyloid disease. In this group, CTS was frequent (37%) and its prevalence correlated with the duration of hemodialysis. In all patients, the abdominal fat tissue, stained with Congo red, was negative for amyloid deposits. These results confirm that CTS is a frequent complication of long-term hemodialysis; however, in this study, no detectable amyloid deposits were found in abdominal subcutaneous fat tissue. Thus, abdominal fat aspiration may not be a reliable screening test for hemodialysis-associated amyloidosis. (<i>Arch Intern Med</i>1987;147:1455-1457)

下顎に発生した脈りゅう性骨嚢胞の１例

The aneurysmal bone cyst is an uncommon bone lesion in the jaws. This lesion is a solitary cystic lesion of bone and was first described as a distinct clinicopathologic entity by Jeffe and Lichtenstein in 1942.We experienced a case of aneurysmal bone cyst of the right mandible in an 11-year-old girl. In April, 1983 she received a blood transfusion to treat a massive hemorrhage that arose from the gingiva at 7 of the mandible. Her course of recovery was without incident and no abnormal bleeding was seen. On April, 1984 she was admitted to The Dental Department of The Hakodate Municipal Hospital because of massive hemorrhage of the gingiva of Ti of the mandible. Roentogenography findings revealed a pear sharped radiolucent area between roots 6 and 7. Angiographical findings showed a coincidence with this roentogenography image and aggregation of contrast medium. We suspected the lesion of blood vessels, and embolization of the maxillary artery was conducted using gelform, but haemostasis was not obtained. After ligation of the right external carotid artery, fenestration of the 7-4 buccal cortid bone of the mandible was conducted.The lesion extended widely from 76 alveolar crest to the diaphysis of the mandible, and a bone cavity of the little finger in size wase found at this site, a large artery branching from the inferior alveolar artery was seen. The extraction of 76 and curettage of the affected site was conducted. On August 3 rd she was admitted to Higashi-Nippon-Gakuen University Hospital for detailed examination and treatment.Roentogenography findings of the 5 site showed a multiloculer radiolucent area of the mesial bone of the mandible. The radiolucent of the roentogenography coincided with the angiographic image of the left externar carotid artery. On August 10, a marginal resection of the mandible was conducted, and the histopathologic findings revealed an cvidence which led to the diagnosis of anrurysmal bone cyst.At present alter a two year lapse nn signs of recuriance wrre seen.

Carpal Tunnel Syndrome With Cystic Bone Lesions Secondary to Amyloidosis in Chronic Hemodialysis Patients

Carpal tunnel syndrome (CTS) is increasingly recognized in patients undergoing chronic hemodialysis. Although the etiology remains obscure, angioaccess-related vascular engorgement and edema, and ischemic neuropathy are two likely possibilities. Amyloidosis is a relatively rare cause of CTS and had previously been thought to occur almost exclusively in patients with multiple myeloma. We report seven patients on chronic hemodialysis who developed CTS and were shown to have amyloid deposition within the synovium. Amyloid was demonstrated by characteristic staining with Congo red on polarizing microscopy and confirmed by electron microscopy. Six patients also exhibited radiolucent carpal bone cysts which were histologically shown to be due to bone replacement by an amyloid-positive synovitis. The average age of the patient and time on dialysis were 59.1 and 7.9 years, respectively. Serum and urine immunoelectrophoresis and bone marrow aspirates showed no evidence for plasma cell dyscrasia in six patients, while one patient did manifest a monoclonal IgA spike. Autopsies in three patients and liver biopsy in another showed no other evidence for disseminated amyloid. These hemodialysis patients exhibited a unique syndrome of CTS, lytic lesions of the carpal bones, and amyloid deposition in the synovium and within the bone cysts.