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At least 235 records · Page 13Linked to original sources
PubMed1989

Decreased production of interleukin-1 by monocytes from patients with lipoid nephrosis.

In order to further characterize monocyte function in patients with lipoid nephrosis (LN), we studied the production of interleukin-1 (IL-1). In this study we examined the ability of peripheral blood monocytes (PBM) from LN patients to produce this monokine in vitro under the stimulus of bacterial lipopolysaccharide (LPS). The levels of IL-1 were decreased in patients with LN compared with those in normal controls and lower in LN patients with nephrotic syndrome (NS) than in those without NS. In contrast, the values in IgA nephropathy (IgAN) patients with or without NS did not differ from normal subjects. The addition of indomethacin, an inhibitor of prostaglandin synthesis, partially restored this defect. These results suggest that the impaired IL-1 production of LN PBM is probably attributable, at least in part, to increased prostaglandin production and possibly influences the immune status of LN patients.

Adolescent↗
PubMedDate not supplied

Predominantly cerebral manifestation in Urbach-Wiethe's syndrome (lipoid proteinosis cutis et mucosae): a clinical and pathomorphological study.

The neurological and psychiatric manifestations in a case of Urbach-Wiethe's disease are reported. The present case is characterized by the predominance of cerebral symptoms: ataxia, psychomotoric seizures, paranoic hallucinatory psychosis and attacks of rage. At autopsy, deposits of amyloid-like material in the walls of the brain vessels could be observed. This finding, together with the mucocutaneous alterations, confirmed the diagnosis of the rare disease know as "lipoid proteinosis cutis et mucosae".

Adult↗
PubMed1985

Depression of local graft-versus-host reaction in patients with lipoid nephrosis.

The local graft-versus-host reaction (GVHR) following intradermal injection of peripheral blood lymphocytes (PBL) in Lewis rats was examined in order to test the cell-mediated immunity (CMI) in 31 patients with renal disease and in healthy controls. It was found that patients with lipoid nephrosis (LN) in the nephrotic stage and systemic lupus erythematosus (SLE) had less mean lesion of the local GVHR and a smaller proportion with positive reactions as compared to normal subjects. This abnormality in LN patients was correlated well with the degree of delayed hypersensitivity skin test responses to purified protein derivative (PPD) and improved with the onset of clinical remission. In contrast, the mean lesion in PBL from LN patients without nephrotic syndrome (NS) and from chronic mesangial proliferative glomerulonephritis (CGN) did not differ from the normal subjects. These findings raise the question of a selective defect in CMI in LN patients with NS. Our studies also indicate that the local GVHR represents a rapid, clinically useful test for assessing cellular immunocompetence in renal disease.

Adolescent↗
PubMed1985

Impaired T-lymphocyte colony formation in lipoid nephrosis.

The T-colony-forming capacity was examined in 13 normal subjects and 14 patients with biopsy-proven lipoid nephrosis (LN). Eighteen additional patients with other well-defined forms of glomerulonephritis were studied as a disease control. Significantly fewer T lymphocyte colonies were found in LN patients and the nephrotic syndrome (NS) than in normal controls. A similar change could be observed in the groups with other types of NS. LN patients with NS had the lowest values of T-colony-forming cells (TCFC), but there was no statistically significant difference between the groups of nephrotic subjects. It was also of interest that peripheral blood lymphocytes (PBL) from 3 patients with lupus nephritis (SLE) produced significantly low levels of TCFC. The lower levels of TCFC in LN could be enhanced when exogenous interleukin 2 (IL 2) was added to the system. We also examined the production of T-colony-stimulating factor (TCSF). The TCSF production by stimulated PBL from LN patients was lower than in normal subjects. To explain our observations, we presumed that the T colony dysfunction seen in LN patients with NS might in part be due to the decreased TCSF activity.

Adolescent↗
PubMed1984

Lipoid proteinosis. A case report.

A 31-year-old Coloured man was admitted to Tygerberg Hospital in 1981 with hoarseness, hyperkeratotic skin lesions and nodules on the eyelids. There was a history of an episode of loss of consciousness. Skull radiographs demonstrated bilateral symmetrical calcifications in the temporal region. Skin biopsy was consistent with a diagnosis of lipoid proteinosis.

Adult↗
PubMed1980

Lipoid nephrosis appearing as acute oliguric renal failure.

Acute oliguric renal failure previously was reported to develop in patients with preexisting idiopathic nephrotic syndrome in association with clinical evidence of vascular volume depletion. We describe an 81-year-old man without recent proteinuria or evidence of preexisting nephrotic syndrome in whom acute oliguric renal failure developed. Renal biopsy disclosed minimal change disease. Nephrotic range proteinuria without severe hypoalbuminemia was detected during the 25-day course of oliguric renal failure. Renal vein thrombosis was excluded. Urine sodium concentration and fractional sodium excretion were reduced, yet left ventricular filling pressure was not subnormal and could be increased to supernormal levels without improvement in glomerular filtration rate. Oliguria and azotemia were corrected following initiation of glucocorticoid therapy. This case suggests that lipoid nephrosis can appear as acute oliguric renal failure without historical or physical evidence of preexisting nephrotic syndrome.

Acute Disease↗
PubMed1978

Lipoid proteinosis; a clinical, pathological and genetic study.

The clinical, pathological, and genetic findings in two closely related families in which a number of cases of lipoid proteinosis occurred are described. The necropsy findings, particularly the neuropathological aspects, in a patient who died from a coincidental pancreatic carcinoma are detailed. The genetic aspects are reviewed.

Aged↗
PubMed1993

[Lipoid proteinosis in 2 sisters].

Lipoid proteinosis was diagnosed in two daughters of a consanguinous marriage on the basis of genetic, clinical, light microscopic and ultrastructural findings. Hyaline material accumulation, thickening of the basal laminae and the resulting typical onion skin phenomenon were observed. In addition to the pathognomonic cutaneous mucosal findings, unusual manifestations such as persistence of deciduous teeth (in one case), oligodontia and intracerebral calcifications were observed. In one patient, the intracerebral calcifications caused epileptic seizures.

Adolescent↗
PubMed1995

Lipoid proteinosis: clinical, histologic, and ultrastructural investigations.

The case of a 12-year-old boy with lipoid proteinosis is reported. Physical examination revealed long-standing varicella-like scars and areas of hyperpigmentation on the face and upper limbs with no evidence of photosensitivity, hoarseness, small papules along the free margins of eyelids, tongue firmness with short frenulum, and widespread papular lesions of the oral cavity. Histologic and ultrastructural examination revealed the characteristic skin changes: pink, hyaline-like, strongly periodic acid-Schiff-positive material in the dermis, surrounding blood vessels, and sweat glands; thin (30 to 35 nm) collagen fibrils interspersed in abundant amorphous material; blood vessels surrounded by thickened, multilayered basement membranes, in which layers of typical, homogeneous basement membrane material were alternating with electronlucent areas filled by various amounts of thin, cross-striated fibrils, arranged perpendicularly. These findings are of great interest since they show a complex relationship between type IV and type III-like collagen components.

Child↗
PubMed1995

[Lipoid proteinosis].

We reported a case of lipoid proteinosis with severe respiratory symptomatology. Diagnosis has been made out of clinical and histological data by laryngeal biopsy. Immunohistochemistry confirms the altered composition of extracellular matrix.

Adult↗
PubMed1998

A novel frameshift mutation 840delA and a novel polymorphism D203A in the steroidogenic acute regulatory protein gene in a Japanese patient with congenital lipoid adrenal hyperplasia. Mutations in brief no. 117. Online.

Congenital lipoid adrenal hyperplasia (CLAH) is an autosomalrecessive disorder characterized by impaired production of all steroids including glucocorticoids, mineralocorticoids and sexsteriods. It has recently been reported that mutations in the steriodogenic acute regulatory protein (StAR) gene cause CLAH. We analyzed the StAR gene in a Japanese patient with CLAH. The patient was revealed to be a compound heterozygote bearing a nonsense mutation Q258X, changing codon 258 (CAG) encoding Gln to the stop codon TAG, and a novel framshift mutation 840delA resulting from deletion of one of the three adenosines normally present in codon 238 (AAA), thus leading to a frameshift after codon 237 (Thr) in the StAR gene. The patient was also revealed to be homozygous for a novel missense point mutation D203A, changing codon 203 (GAC) encoding Asp to GCC encoding Ala in the StAR gene. To elucidate the significance of the D203A mutation, we analyzed the StAR gene sequence in twenty normal subjects, and found that all of them were homozygous for the D203A mutation, indicating that the D203A mutation is an innocent polymorphism. In conclusion, we have identified a novel frameshift mutation 840delA which seems to cause 840delA and the first polymorphism D203A in the human StAR gene.

Adrenal Hyperplasia, Congenital↗
PubMed1997

Mineral oil lipoid pneumonia in a child with anoxic encephalopathy: treatment by whole lung lavage.

We describe a case of exogenous lipoid pneumonia in a child with anoxic encephalopathy who was taking mineral oil for constipation. Computed tomography produced images suggesting this condition, and the diagnosis was confirmed by demonstrating the presence of lipid-laden alveolar macrophages in the bronchoalveolar lavage fluid. Despite discontinuing the offending agent, the pulmonary infiltrates did not improve; however, successful resolution was obtained by whole lung lavage.

Bronchoalveolar Lavage↗
PubMed1981

Lipoid pneumonia caused by oil mist exposure from a steel rolling tandem mill.

Five of nine active tandem mill operators exposed at work to aerosolized hydrocarbon mist were referred for evaluation of respiratory complaints. The worker with the longest exposure had reduced lung volumes; he was admitted to the hospital for detailed study. Exercise studies revealed work load limited by ventilation and arterial oxygen desaturation. Flexible fiberoptic bronchoscopy with bronchoalveolar lavage and transbronchial biopsy revealed evidence of lipoid pneumonia. Assessment of the mill revealed levels of respirable oil mist by personal samplers throughout the area far below the currently accepted standard of 5 mg/M3. These findings confirm a 20-year-old hypothesis of J.G. Jones regarding the hazard of oil mist in this industrial setting.

Adult↗
PubMed1989

Bronchoalveolar lavage in the diagnosis of lipoid pneumonia.

Lipoid pneumonia (LP) is an uncommon entity with the characteristic radiographic features and histologic findings of alveoli filled with vacuolated, lipid-laden histiocytes. We questioned whether bronchoalveolar lavage (BAL) could be useful in the confirmation of LP. We examined lipid stains (oil-red O) in BAL specimens from 18 cases, representing a variety of pulmonary disease states, and compared them with an index case of confirmed LP. The index case of LP had a history of chronic intranasal use of mentholated petrolatum with subsequent x-ray findings of progressive air bronchograms. Positive histochemical confirmation (oil-red O) performed on frozen sections of transbronchial lung biopsy was obtained. Eleven of the non-LP cases had no intracellular staining of BAL macrophages with oil-red O stain, whereas the index case of LP exhibited markedly positive intracytoplasmic staining of macrophages for lipid. The remaining seven cases showed minimal to mild lipid staining with only one other case having moderate staining. This study demonstrates that BAL macrophages from patients with a variety of pulmonary states and without suspected LP do not demonstrate significant staining for intracellular lipids, in contrast with the expected strong positivity of LP. Lipid staining of BAL specimens, although not entirely specific, may be a preferred method for confirming the diagnosis of LP, thereby avoiding more invasive procedures.

Adult↗
PubMed1980

High-pressure liquid chromatographic analysis of pramoxine hydrochloride in high lipoid aerosol foam dosage form.

A rapid and quantitative method for the determination of pramoxine hydrochloride by high-pressure liquid chromatography is presented. The drug is extracted as the salt from a preparation with a high lipoid composition by partitioning it to the aqueous phase of an ether-methanol-water-acetic acid system. The extract is chromatographed on an octadecylsilane bonded packing with a methanol-water-acetic acid-methanesulfonic acid mobile phase. The time required for each separation is approximately 6 min. Analytical recoveries of 100.4 +/- 1.5% were obtained.

Aerosols↗
PubMed2003

Seasonal variation of the lipoidal matters and hypolipidaemic activity of the red alga Corallina officinalis L.

The lipoidal matters of Corallina officinalis L. showed a seasonal nonsignificant quantitative variation. However, the fatty acids revealed a relative increase in the summer and winter, while unsaponifiable matter exhibited a slight increase in the spring. The GC/MS analysis of saponifiable and unsaponifiable matter of the algal samples collected in different seasons revealed that samples collected in the spring contained a low cholesterol content and high steroidal compounds as well as high polyunsaturated fatty acids. The alcohol extract, hexane extract and fatty acid fraction of this algal sample exhibited a significant hypolipidaemic activity. Also, two biologically active fractions of hydrocarbons were isolated by CC technique from the hexane fraction of C. officinalis L. and identified by GC/MS.

Animals↗
PubMed1993

Cushing's syndrome secondary to malignant lipoid cell tumor of the ovary.

Malignant lipoid cell tumors of the ovary are rare lesions that are frequently associated with endocrinologic abnormalities. A case of a woman with this lesion who developed Cushing's syndrome with progression of tumor is presented. Neither aggressive medical therapy with ketoconazole nor multiagent chemotherapy was beneficial in controlling tumor growth or physical and biochemical manifestations of Cushing's syndrome.

Aged↗