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PubMed1996

Diagnostic value of endomyocardial biopsies of the right ventricular septum in arrhythmias originating from the right ventricle.

The aim of the study was to classify the value of endomyocardial biopsies taken from the right ventricular septum in patients with ventricular tachyarrhythmias originating from the right ventricle. In a cohort of 62 young patients (mean age 35.6 years) 4-5 endomyocardial biopsies were taken from the right ventricular septum and myocardial atrophy, fibrosis and lipomatosis were analysed qualitatively and semi-quantitatively. Much interest was focused on the arrangement and type of fibrosis. Lipomatosis, myocardial atrophy and a finer form of fibrosis surrounding individual myocytes or groups of myocytes were judged as the typical histopathological findings of arrhythmogenic right ventricular dysplasia. The cohort of patients was subdivided clinically into idiopathic right ventricular outflow tract tachycardia (n = 50) and arrhythmogenic right ventricular dysplasia (n = 12). In the group of arrhythmogenic right ventricular dysplasia pathological findings were found in 92% with typical fibrolipomatosis in two cases, severe fibrosis ( > 40% per biopsy) in 5 cases, a finer form of fibrosis surrounding individual myocytes without lipomatosis in two cases and a slight interstitial or subendocardial fibrosis in two cases. In only one patient the endomyocardial biopsy was normal. In right ventricular outflow tract tachycardia normal endomyocardial biopsy findings predominated (80%). In two cases lipomatosis without fibrosis was a more or less normal finding, and in two other cases a slight interstitial or subendocardial fibrosis was found. Fibrosis was judged to be moderate or severe with discrete signs of inflammation in only two cases; in a long-term follow-up these two patients developed typical features of idiopathic dilated cardiomyopathy. Endomyocardial biopsies taken exclusively from the right ventricular septum were able to distinguish between right ventricular outflow tract tachycardia and arrhythmogenic right ventricular dysplasia, although the typical finding of fibrolipomatosis in cases with arrhythmogenic right ventricular dysplasia was rare. Characteristics of fibrosis are the key to the correct diagnosis provided that histopathological analysis is based on experienced qualitative criteria (type and arrangement of fibrosis). In conclusion, qualitative and semi-quantitative analyses of endomyocardial biopsies are far better than computerized quantitative methods. Biopsies from the free right ventricular wall cannot be generally recommended if a correct definition of arrhythmogenic right ventricular dysplasia in endomyocardial biopsies from the right ventricular septum is used.

Adult↗

PubMed1988

Multiple familial angiolipomatosis: treatment of liposuction.

Lipomas constitute the most common soft-tissue tumors [11] and may occur sporadically or as one of several inherited disorders. Benign fatty tumors can be classified into three major categories: solitary lipomas, familial multiple lipomatosis, and congenital diffuse lipomatosis [4]. The latter has also been referred to as multiple symmetric lipomatosis, Madelung's disease, or Launois-Bensaude syndrome. Within the categories a more vascular varient of the lipoma, the angiolipoma, has been described separately by some authors, but in many reports it is not distinguished from a lipoma. Liposuction has been used for the treatment of solitary lipomas, but to our knowledge liposuction has not previously been reported for excision of angiolipomas or in familial cases involving multiple lipomas as described here.

Adipose Tissue↗

PubMedDate not supplied

Repeated failure of epidural analgesia: an association with epidural fat?

BACKGROUND AND OBJECTIVES: To report the case of a patient who experienced repeated failed epidural analgesia associated with an unusual amount of fat in the epidural space (epidural lipomatosis). CASE REPORT: A 44-year-old female presented for an elective small bowel resection. An L(1-2) epidural catheter was placed for postoperative analgesia. The patient gave no indication of having pain at the time of emergence from general anesthesia or in the first 2 hours in the recovery room. Assessment of the level of hypoesthesia to ice while the patient was comfortable in the recovery room suggested a functional epidural catheter (cephalad level of T(10)). Two hours after admission to the recovery room the patient began to complain of increasing pain. Another 6 mL 0.25% bupivacaine was administered via the catheter. The patient's pain decreased, but remained substantial, and there was minimal evidence of sensory block above the T(10) level. Subsequently, a T(10) epidural catheter was placed. Testing confirmed proper placement of the catheter in the epidural space at the T(10) level. A test dose of 5 mL 0.25% bupivacaine resulted in prompt and complete relief of the patient's pain. However, the level of hypoesthesia to ice did not exceed the T(10) level. Approximately 1 hour later the patient complained of increasing discomfort again. There was no evidence of any sensory block, and there was no response to a bolus of 8 mL 1% lidocaine. A thorough examination of the patient did not suggest any cause for the pain other than a malfunctioning epidural catheter. A third epidural catheter was placed at the T(8-9) level. This catheter was again confirmed to be in the epidural space with a test dose of 10 mL 0.5% bupivacaine. The level of hypoesthesia to ice was restricted to a narrow bilateral band from T(7)-T(9). Analgesia failed 2 hours later. The epidural catheter was removed and the patient's pain was subsequently managed with intravenous patient-controlled analgesia (PCA) morphine. A magnetic resonance imaging (MRI) scan revealed extensive epidural fat dorsal to the spinal cord from C(5)-C(7) and from T(3)-T(9). An imaging diagnosis of asymptomatic epidural lipomatosis was established. CONCLUSION: We have described a case of repeated failure of epidural analgesia in a patient with epidural lipomatosis.

Adipose Tissue↗

PubMed2004

Effectiveness of high trough levels of cyclosporine for 5 months in a case of steroid-dependent nephrotic syndrome with severe steroid toxicity.

Glucocorticoid treatment for steroid-dependent nephrotic syndrome (NS) is associated with severe adverse effects, such as bone fractures and epidural lipomatosis. Furthermore, a high trough level of cyclosporine (CsA) over an extended period of time is known to induce CsA nephropathy. We present a girl with steroid-dependent NS and steroid-induced vertebral compression fractures and epidural lipomatosis who was treated with a high-dose of prednisolone after experiencing several relapses. A high CsA trough level (between 147 and 225 ng/mL) over a period of only 5 months was effective in improving the vertebral compression fractures, alleviating the epidural lipomatosis by enabling the discontinuation of prednisolone treatment. Thus, high trough levels of CsA over a short period of time may enable prednisolone to be discontinued in cases of steroid-dependent NS without causing any clinical, histological, serum and/or urinary CsA-related adverse effects.

Child, Preschool↗

PubMedDate not supplied

From the archives of the AFIP: benign musculoskeletal lipomatous lesions.

Benign lipomatous lesions involving soft tissue are common musculoskeletal masses that are classified into nine distinct diagnoses: lipoma, lipomatosis, lipomatosis of nerve, lipoblastoma or lipoblastomatosis, angiolipoma, myolipoma of soft tissue, chondroid lipoma, spindle cell lipoma and pleomorphic lipoma, and hibernoma. Soft-tissue lipoma accounts for almost 50% of all soft-tissue tumors. Radiologic evaluation is diagnostic in up to 71% of cases. These lesions are identical to subcutaneous fat on computed tomographic (CT) and magnetic resonance (MR) images and may contain thin septa. Lipomatosis represents a diffuse overgrowth of mature fat affecting either subcutaneous tissue, muscle or nerve, and imaging is needed to evaluate lesion extent. Lipoblastoma is a tumor of immature fat occurring in young children, and imaging features may reveal a mixture of fat and nonadipose tissue. Angiolipoma, myolipoma, and chondroid lipoma are rare lipomatous lesions that are infrequently imaged. Spindle cell and pleomorphic lipoma appear as a subcutaneous lipomatous mass in the posterior neck or shoulder, with frequent nonadipose components. Hibernoma appears as a lipomatous mass with serpentine vascular elements. Benign lipomatous lesions affecting bone, joint, or tendon sheath include intraosseous lipoma, parosteal lipoma, liposclerosing myxofibrous tumor, discrete lipoma of joint or tendon sheath, and lipoma arborescens. Intraosseous and parosteal lipoma have a pathognomonic CT or MR appearance, with fat in the marrow space or on the bone surface, respectively. Liposclerosing myxofibrous tumor is a rare intermixed histologic lesion commonly located in the medullary canal of the intertrochanteric femur. Benign lipomatous lesions may occur focally in a joint or tendon sheath or with diffuse villonodular proliferation in the synovium (lipoma arborescens) and are diagnosed based on location and identification of fat. Understanding the spectrum of appearances of the various benign musculoskeletal lipomatous lesions improves radiologic assessment and is vital for optimal patient management.

Adolescent↗

PubMed1997

Symmetric lipomatoses in female patients.

BACKGROUND: Symmetric lipomatoses are characterized by marked symmetric deposition of diffusely distributed fatty tissue. Though relatively common disorders, they are rather rarely reported in the literature, possibly being misdiagnosed as general obesity. While the differential diagnosis of symmetric lipomatosis versus general obesity may not appear difficult in males, it is obviously problematic in females. OBSERVATIONS: We describe the findings in 6 representative female patients with symmetric lipomatoses: 3 with benign (multiple) symmetric lipomatosis and 3 with female zonal obesity. The former disorder was characterized by massive, firm, symmetric fat deposition predominantly around the neck and shoulder girdle and was clearly associated with alcohol abuse and/or liver disease. There were no malignant tumors of the upper airways. In the latter case, fatty tissue had accumulated mainly at the buttocks and thighs but characteristically spared the feet and hands. Tenderness was a common symptom. This disorder showed familial predisposition. Histology in both cases revealed normal fatty tissue which was neither encapsulated nor septally divided. CONCLUSIONS: We suggest that the term 'symmetric lipomatoses' refers to two separate disorders, benign (multiple) symmetric lipomatosis and female zonal obesity.

Adipose Tissue↗

PubMed1981

Lipomatous tumors of the abdominal cavity: CT appearance and pathologic correlation.

Twenty patients with pathologically confirmed extraparenchymal intraabdominal lipomatous tumors, including two lipomas, two cases of diffuse infiltrating lipomatosis, and 16 liposarcomas, were examined by computed tomography (CT). The CT appearance of these tumors closely correlated with their gross and microscopic pathologic anatomy. Distinctive CT features differentiated simple lipomas from diffuse infiltrating lipomatosis and from liposarcomas. The densities of these tumors, including the variable densities of liposarcomas, were explained by their tissue composition. Lipomas, diffuse infiltrating lipomatosis, and lipogenic liposarcomas were predominantly of fat density, whereas myxoid liposarcomas were of a higher range of densities. Liposarcomas often contained more than one type of tumor tissue, resulting in masses of distinctly different densities coexisting within the same tumor. CT accurately depicted the presence, location, and size of the tumors and provided information about their relation to adjacent structures.

Abdominal Neoplasms↗

PubMed1998

[Case report of a large lipomatous soft tissue tumor].

A case of diffuse infiltrating lipomatosis is reported. This entity represents a particular type of lipoma. It can be distinguished from other fatty tumors by its localisation, extent, therapeutic procedure, and prognosis. Magnetic resonance imaging (MRI) enables the differentiation from lipomatosis pelvis and malignant tumors as well. Histopathological examination, however, is necessary to exclude liposarcoma. Further growth or recurrence of diffuse infiltrating lipomatosis can be detected by MRI follow-up examinations.

Buttocks↗

PubMed1996

Magnetic resonance imaging for diagnosis of Shwachman's syndrome.

Shwachman's syndrome is a rare disease characterized by the association of chronic diarrhea due to exocrine pancreatic insufficiency, metaphyseal dysostosis, and neutropenia. The diagnosis requires demonstration of lipomatosis, or fatty replacement of the pancreas, which is the typical pathological feature of the disease. Magnetic resonance imaging (MRI) was performed in 13 patients with exocrine pancreatic insufficiency, 7 with Shwachman's syndrome, 2 with Pearson's syndrome, 1 with normal sweat test later diagnosed as cystic fibrosis, and 3 without identified syndrome, and in 7 control children. Ultrasonography in the patients did not differentiate between atrophy and lipomatosis and could not be performed in 3. MRI visualized the pancreas in all. The same image was noted in all patients with Shwachman's syndrome with a normal-sized or enlarged pancreas, a hyperintense signal on T1- and T2-weighted image, and a null signal on short time inversion recovery (STIR)-weighted image, characteristic of fat. In all other patients, the findings were very different: The pancreas was a small structure surrounded with fat. In 1 patient without identified syndrome, the pancreas appeared to be partially replaced with fat. MRI is an excellent imaging technique to correlate the nature of a tissue and its radiological representation, especially fat, which gives a very typical signal. In our brief series of patients with Shwachman's syndrome, MRI had 100% positive predictive value in demonstrating lipomatosis. In atypical cases of pancreatic insufficiency in which some of the clinical features of Shwachman's syndrome are absent. MRI is an invaluable aid in the diagnostic procedure.

Adolescent↗

PubMed1998

Nevus psiloliparus: a distinct fatty tissue nevus.

BACKGROUND: Encephalocraniocutaneous lipomatosis is usually associated with a peculiar type of fatty tissue nevus which represents a smoothly surfaced and hairless lesion involving the scalp. This disorder has so far not been recognized as a cutaneous entity. OBJECTIVE: The purpose of this article is to describe the characteristic features of this nevus and to give it a name. METHOD: From the study of two cases and from a review of the literature we delineate the clinical and histopathological criteria of this disorder for which we propose the term 'nevus psiloliparus'. This name is derived from the Greek words psilós = hairless and liparós = fatty, and describes the two most characteristic features of the disorder. RESULTS: A comprehensive comparison shows that nevus psiloliparus can be distinguished from other types of fatty tissue nevi by clinical criteria such as localization on the scalp, a flat smooth surface and absence of hair follicles, by the histopathological feature of isolated arrector pili muscles and by the presence of associated extracutaneous features in the form of encephalocraniocutaneous lipomatosis. In particular, nevus psiloliparus can be separated from the Hoffmann-Zurhelle nevus that has so far never been observed in cases of encephalocraniocutaneous lipomatosis. CONCLUSION: The presently available data suggest that nevus psiloliparus represents a distinct cutaneous entity. Future clinical and genetic research should show whether this concept holds true.

Adipose Tissue↗

PubMed1995

[Posterior epidural adipose tissue and the narrow lumbar canal: replacement tissue or cause of impingement?].

The role played by the epidural fat has been reported in lipomatosis induced by exogenous glucocorticoids and in severe obesity with lipomatosis. The role played by the "normal" posterior epidural fat (PEF) in lumbar canal stenosis (LCS) is less well known. The purpose of this study was to determine the part taken by PEF in LCS patients without endocrine disease, corticosteroid therapy or obesity. For this, we tried to specify the amount and distribution of PEF among the soft tissues in the vertebral canal, to demonstrate the involvement of PEF in dural sac compression, to describe the radiological features observed in cases of LCS and to look for associated morphological factors. The records of 30 LCS patients without exogenous or endogenous lipomatosis and in whom the essential pathogenic factor in 40 levels was PEF were reviewed retrospectively. At disc level, PEF was evaluated in the lower part of the mobile segment by means of CT or MRI axial sections cut through one or two spaces between L2-L3 and L4-L5. Measurements were made in 25 men (80%) and 6 women (20%) aged from 33 to 83 years (mean: 58 years). Most patients were suffering from lumbar pain, radiculopathy and/or neurogenic intermittent claudication. The data measured were: antero-posterior (AP) diameter of the dural sac, AP diameter of the bony lumbar canal (BLC), interligamentous distance (ILD) opposite the articular facets, and surface of PEF. The soft elements present on the midline--anterior epidural space (AES) and posterior epidural (PEF)--were expressed as percentage of the AP diameter of the bony lumbar canal.(ABSTRACT TRUNCATED AT 250 WORDS)

Adipose Tissue↗

PubMed2000

Uncoupling protein-1 mRNA expression in lipomas from patients bearing pathogenic mitochondrial DNA mutations.

Multiple symmetric lipomatosis (MSL) is a rare disorder characterised by large subcutaneous fat masses in some parts of the trunk. Mitochondrial disfunction is common in MSL, but the identity of the adipose cells developing in multiple lipomas is not well known. We determined that brown adipose tissue-specific uncoupling protein-1 (UCP-1) mRNA is expressed in the lipoma of a multiple symmetric lipomatosis patient bearing the 8344 mutation in the tRNALys gene of mitochondrial DNA. UCP1 mRNA was not detected in normal subcutaneous fat from the same patient or in the lipoma of another patient bearing a different mutation in the same tRNALys gene. These findings implicate brown adipose cells as the origin of lipomas in a subset of patients bearing tRNALys mutations in mitochondrial DNA.

Adipose Tissue, Brown↗

PubMed1994

Magnetic resonance imaging of skeletal muscles in idiopathic inflammatory myopathies of adults.

The purpose of the study was to describe typical MRI findings in various types of idiopathic inflammatory myopathies in adulthood and to correlate the MRI with histopathological and electromyographic findings, and the serum creatine kinase (CK) activity. A third goal was to assess the diagnostic value of the use of gadolinium-DTPA (Gd-DTPA). Fifty-eight patients (35 women, 23 men), aged 21-83 years (median age 59 years), suffering from idiopathic myositides (13 with acute and 45 chronic diseases; 25 with polymyositis, 14 with dermatomyositis, 8 with granulomatous and 11 with inclusion body myositides) were examined with MRI. Seventeen of them received an intravenous infusion of Gd-DTPA. Histopathological and MRI findings of 21 muscles of 18 patients were compared. MRI of skeletal muscles showed abnormal signal intensities in 56 (96.6%) of the 58 patients. MRI abnormalities were found more often than elevated CK activity (P < 0.001). The hyperintensity of T2-weighted images was more conspicuous than on T1-weighted images in 26 (44.8%) patients, indicating oedema-like abnormalities. MRI of 50 (86.2%) patients showed fat replacement. In acute myositides, oedema-like abnormalities were more often visible and in muscle lipomatosis less often visible than in chronic diseases (P < 0.05 each). In dermatomyositis oedema-like abnormalities were more and lipomatosis less frequent than in the other types of myositis (P < 0.005) and correlated with the acuteness of the disease.(ABSTRACT TRUNCATED AT 250 WORDS)

Adult↗

PubMed2003

[Surgical therapy of Madelung's disease in the head and neck area].

PATIENTS AND METHODS: Between 1995 and 2001 we treated 12 patients with benign symmetric lipomatosis in the head and neck. The group comprised 11 men and 1 woman aged 34-62 (M=47). The most frequent complaints were diminished range of motion of the head and obstruction when eating or speaking. Combined lipectomy and liposuction were performed in all patients, with liposuction being carried out in a second session. RESULTS: The mean follow-up period was 3 years. The functional results were very satisfactory in all patients. Of the 12 patients, 9 were also satisfied with the aesthetic outcome.A recurrence occurred in three patients 1.5, 2, or 3 years after the operation. Severe complications were not observed. CONCLUSIONS: According to our experience, combined lipectomy and liposuction represents a successful procedure in treating benign symmetric lipomatosis in the head and neck region.Nevertheless, advantages and drawbacks of the two techniques should be equally considered prior to surgery.

Adult↗

PubMed2003

Fibrolipomatous hamartoma in the foot: atypical MR imaging findings.

Lipomatosis of a nerve is a well-known but uncommon entity mostly seen in the median nerve. Magnetic resonance imaging (MRI) has been shown to provide pathognomonic features, obviating the need for diagnostic biopsy. We present a case of lipomatosis of a branch of the medial plantar nerve with an atypical appearance on magnetic resonance imaging.

Adult↗

PubMedDate not supplied

Scrotal involvement in Madelung disease: clinical, ultrasound and MR findings.

Multiple symmetrical lipomatosis, also known as Madelung disease, Launois-Bensaude syndrome, and benign symmetrical lipomatosis, is an uncommon disease that is characterized by the presence of multiple and symmetrical deposits of nonencapsulated adipose tissue in the neck, upper trunk, arms, and legs. It affects mainly men, with a higher incidence in the Mediterranean area, and is typically associated with high alcohol consumption and a high prevalence of peripheral neuropathy that usually appears long after the development of lipomatous masses. We report the case of a patient with involvement of the scrotum. We present clinical, ultrasound, and magnetic resonance appearances of these scrotal lipomatous masses for the first time.

Adult↗

PubMedDate not supplied

Lipomatous disorder of the peri-trochanteric soft tissue: case report and review.

Disorders of fatty tissue metabolism and body contour are frequently described in the literature. Several different syndromes have been ascribed to recurring characteristics of fatty deposition with similar underlying etiologies. These syndromes can generally be classified into the lipomatoses (abnormal accumulations of fat) and the lipodystrophies (abnormal fat atrophy). The authors describe a case of isolated symmetrical lipomatosis of the soft tissue overlying the trochanters that developed a recurrence after treatment with liposuction. They have attempted to match this patient's disorder into 1 of several reported syndromes, with benign symmetric lipomatosis and acquired partial lipodystrophy at the top of the differential diagnosis. However, some characteristics of this patient may warrant a separate categorization for her condition. In this case report, the current literature on disorders of fatty tissue metabolism is reviewed and a discussion of relevant factors and issues surrounding their clinical significance and management is provided.

Adult↗

PubMed2003

Proptosis after retrobulbar corticosteroid injections.

PURPOSE: This report describes the clinical, radiographic, and histopathologic findings in patients with proptosis secondary to lipomatosis after retrobulbar corticosteroid injection. DESIGN: Retrospective, noncomparative, interventional case series and review of the literature. METHODS: Five patients who developed symptomatic unilateral proptosis after steroid injection were studied and the literature was reviewed. RESULTS: No previous description was found in the literature. All five cases were studied with computed tomography, and two cases were confirmed with histopathology. No fibrosis or granulomatous inflammation was identified. CONCLUSIONS: Orbital lipomatosis is a potential complication of retrobulbar steroid injections. Symptomatic relief can be provided by a transconjunctival approach to the lower lid fat compartment.

Adult↗