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PubMed1976

Human infection with Dermatophilus congolensis.

Dermatophilosis is a skin disease in animals and humans caused by the actinomycete Dermatophilus congolensis. This microorganism causes the skin disease in sheep commonly referred to in Australia as "lumpy wool" or mycotic dermatitis. One proven case of human dermatophilosis and two cases with features which are clinically highly suggestive of the disease from South Australia are described. There has been only one previous report of human infection caused by D. congolensis, which was from the United States in 1961. A brief account of the history, characteristics and life cycle of D. congolensis is given. Human infection in Australia may be not uncommon. A diagnosis can be made by direct microscopy of scab material from the lesions. For this purpose, dry scabs should be sent to the laboratory.

Actinomycetales↗

PubMed1981

The "lumpy-bumpy" elastic fiber. A marker for long-term administration of penicillamine.

A 29-year-old woman with Wilson's disease developed dermolytic skin lesions 2 years after initiation of treatment with penicillamine. Eight years later, still on penicillamine therapy, striae appeared over both of her breasts. Biopsy of involved skin during the 10th year of treatment with penicillamine revealed characteristic lumpy-bumpy alterations of dermal elastic fibers which were not present in the first skin biopsy 8 years previously. Biopsy of a stria showed changes similar to those in the dermolytic skin lesions. Lumpy-bumpy elastic fibers are pathognomonic for penicillamine-induced elastosis. They are easily recognizable with examination by conventional microscopy. Their appearance may serve as a warning of potentially serious, widespread elastic tissue involvement. These abnormal elastic fibers are not only found in the skin, but also in the lungs.

Adult↗

PubMed1997

Use of degenerate primers and heat-soaked polymerase chain reaction (PCR) to clone a serine protease antigen from Dermatophilus congolensis.

Serine proteases are thought to be involved in the initial attack on sheep skin by Dermatophilus congolensis and are obvious antigens for inclusion in a vaccine to prevent lumpy wool disease (dermatophilosis). Degenerate primers were designed after alignment of seven bacterial serine proteases. Inosine was incorporated into the primers at positions of three- and four-base redundancy, and this reduced the complexity of the primer mixtures from several thousand to sixteen different sequences for each primer. The primers were validated by production and sequencing of amplicons from serine protease genes in Bacillus subtilis and Serratia marcescens. The primers were used with heat-soaked polymerase chain reaction (PCR) to produce amplicons from two D. congolensis strains, AG and MB. In the amplicon codons for arginine, rather than the expected serine, were found where inosine was used for both the first and third positions for a codon in the primer. A search with the deduced amino acid sequences of the amplicons showed significant similarity to a keratinase and other serine proteases from various organisms. Similarity was most apparent around the active site residues and other essential secondary structural elements.

Actinomycetales↗

PubMed1986

Penicillamine-induced elastosis perforans serpiginosa. Tip of the iceberg?

Elastosis perforans serpiginosa (EPS) is now a well-recognized potential complication of long-term penicillamine therapy. By itself, EPS appears to be a relatively innocuous cutaneous side effect of penicillamine. However, suspicion has been raised in recent literature that EPS may represent only a superficial manifestation of more serious penicillamine-induced systemic elastic tissue damage, particularly involving blood vessels. This is a report of a patient with Wilson's disease who was treated with penicillamine for 14 years. She developed EPS, and histologic examination of the skin revealed the characteristic penicillamine-induced "lumpy-bumpy" elastic fibers in the dermis. More important, nonlesional skin showed the same elastic fiber changes. Of greatest significance was the finding of identical elastic fiber alterations in an artery.

Adult↗

PubMed1979

"Lumpy-bumpy" elastic fibers in the skin and lungs of a patient with a penicillamine-induced elastosis perforans serpiginosa.

Penicillamine-induced cutaneous elastosis perforans serpiginosa associated with a large air-cyst in the right lung is described in a 29-year-old female patient with Wilson disease. Identical light and electron-microscopic changes were present in both dermal and pulmonary elastic tissue, suggesting a disseminated drug-induced cutaneo-visceral elastosis. Lung cysts have not been previously reported in association with long term penicillamine treatment. The electron-microscopic morphology of the elastic fibers was found to be "specific" enough to allow separation of penicillamine-induced elastosis perforans serpiginosa from other forms of this disease.

Adult↗

PubMed1999

The management of women with breast symptoms referred to secondary care clinics in Sheffield: implications for improving local services.

Information was collected about 302 women referred for breast symptoms and seen in surgical outpatient or outreach clinics during one month at two hospitals in Sheffield. Three-quarters of the women (n = 244) were referred to specialist breast clinics, 22% (n = 70) were referred to general surgical clinics and 3% (n = 6) were referred to outreach clinics. The ages of the women ranged from 16 to 85 years with a mean and median age of 45 years. Some 200 women (66%) presented with a lump or lumpiness, 42 women (14%) presented with pain, 29 women (10%) had a skin and/or nipple problem, and the remaining 31 women (10%) were concerned about their family history or reported other symptoms. A total of 23 women (8%) were diagnosed as having cancer, 180 (60%) were diagnosed as having benign breast disease, and 99 (33%) were diagnosed as normal. Of the 23 women with cancer, 22 were over 40 years of age; 21 women presented with a lump, one presented with pain, and one presented with metastatic disease. The time required to reach a final clinical diagnosis varied from the same day as the clinic visit to 35 weeks, with a median time of 3 weeks. Surgeons assessed the appropriateness of GPs' referrals for 257 cases and judged that 122 (47%) could have been managed by a GP. The implications of the findings for the organisation of specialist outpatient clinics are discussed, and a categorisation of women as either urgent or routine cases is suggested.

Adolescent↗

PubMed2003

Weber Christian disease.

Weber Christian disease or idiopathic lobular panniculitis is a skin condition that features recurring inflammation in the fat layer of the skin. We report a case of a 17 year old boy with Weber Christian disease as its occurence is extremely rare with only sparse case reports available, who presented with eighteen-month history off fever with chills off and on, arthralgia predominantly of the knees, ankles, wrists and elbows without swelling or gross limitation of movement. There was also swelling around the eyes and puffiness of the face with lumpy swellings on the jaw, cheeks and chin and painful lumps on the anterior abdominal wall. On examination a febrile patient with pallor, pitting edema of the legs, periorbital edema and multiple non-matted firm to hard tender swellings of the face, jaw and chin and multiple firm tender lumps in the anterior abdominal wall was noted. Investigations revealed anemia, leucopenia, raised sedimentation rate and mild rise of liver enzymes. Biopsy of the abdominal nodule demonstrated a lobular panniculitis without vasculitis as seen in Weber Christian disease.

Adolescent↗

PubMed1992

Pseudoxanthoma-elasticum-like skin changes induced by penicillamine.

The interference of penicillamine with collagen and elastin cross-linking can lead to wrinkling and anetoderma-like lesions in flexural areas as well as fragility and hemorrhagic blisters in pressure areas. These changes are seen primarily in patients with Wilson's disease or cystinuria who are on long-term therapy. This is a report of a patient with cystinuria on long-term, high-dose penicillamine who developed pseudoxanthoma-elasticum-like lesions. Coalescent yellow papules with a 'plucked-chicken skin' appearance were seen in the axillae and on the neck while redundant skin folds were noted in the anterior axillary line and lower buttocks. By light and electron microscopy, involved and uninvolved skin demonstrated 'lumpy-bumpy' dermal elastic fibers with no calcium deposition. These histologic changes are similar to those previously described in patients with penicillamine-induced skin lesions.

Aged↗

PubMed1994

Mid dermal elastolysis: case report and review of the literature.

A new case of typical mid dermal elastolysis was studied with electron microscopy. Elastic fibers were irregularly branched or lumpy, but subcomponents of these fibers were often normal. In some fibers, loose assembly of skeleton fibrils and aggregation of dense substances were observed. Phagocytosis of normal and abnormal elastic fibers by dermal macrophages was observed.

Adult↗

PubMed2005

Childhood Henoch-Schönlein purpura nephritis and IgA nephropathy: one disease entity?--A clinico-pathologically comparative study.

In order to characterize their relationship through clinicopathological comparison between IgA nephropathy and Henoch-Schönlein purpura nephritis (HSPN), 31 children with IgA nephropathy aged between 3 to 15 years and 120 children with HSPN aged between 4 to 15 years were compared with each other in clinical manifestation, blood biochemistry, serum immunology and follow-up study. Renal pathological findings under light microscope, immunofluorescence and electronic microscope were analyzed and also compared between 31 children with IgA nephropathy and 32 biopsied children with HSPN. The results showed that the onset age was over 12 years in 25.8% children with IgA nephropathy, but only 10% in HSPN (P < 0.05). The clinical patterns of IgA nephropathy and HSPN were similar, but extra-renal manifestations were more often in HSPN, all of them had skin purpura, 59% had gastrointestinal symptoms and 47% suffered from arthralgia, compared with only abdominal pain in 3.2% children with IgA nephropathy. The renal pathological investigation showed global sclerosis in 35.5% of IgA nephropathy and 3.1% of HSPN, mesangial sclerosis in 41.9% of IgA nephropathy and 6.3% of HSPN, but endothelial proliferation in 65.6% of HSPN and 29% of IgA nephropathy (all P < 0.01). Thin basement membrane nephropathy was only found in 6.5% children with IgA nephropathy, no in HSPN. The electronic dense deposits in HSPN were sparse, loose and wildly spread in glomerular mesangium, subendothelial area and even intra basement membrane, but it was dense, lumpy and mostly limited in mesangium and paramesangium in IgA nephropathy. Predominant IgA deposits were found in 81.2% of HSPN, and overwhelming IgG deposits in 12.5% of HSPN with relatively weak IgA deposits, moreover 6.3% of HSPN showed linear IgG deposits in glomerular capillary. Totally 71.9% of HSPN had IgG deposits in glomeruli and only 19.4% of IgA nephropathy showed glomerular IgG deposits (P < 0.01). No IgG deposit was observed in 81.6% of IgA nephropathy, among them most showed IgA and IgM and/or C3 deposits, moreover overwhelming IgG deposits and linear IgG deposits couldn't be found in IgA nephropathy. Mean 20 months follow-up showed complete remission in 72.5% of HSPN, but only 19.4% in IgA nephropathy after 34 months follow-up. Moreover, 64.5% of IgA nephropathy had consistent hematuria and proteinuria and 16.1% had active nephritides (P < 0.05). It was concluded that significant clinico-pathological difference was found between HSPN and IgA nephropathy, which didn't support the one disease entity hypothesis. HSPN and IgA nephropathy are probably two diseases with similar immune abnormalities.

Adolescent↗

PubMed1977

Modified techniques for the isolation of Dermatophilus spp. from infected material.

Two improved techniques are described for the isolation of Dermatophilus spp. The first is suitable for samples of scab from lesions of the disease in animals, except for sheep affected with lumpy wool. The second is for the isolation of the organism from samples of lumpy wool in sheep. Both techniques are simple, rapid and reliable and are based on releasing the organism by pulverizing the scab and then by suppressing contaminants by including polymixin B sulphate in the medium. Excellent results are obtained with or without pre-incubation exposure to CO2.

Actinomycetales↗

PubMed2003

[A clinico-pathological study comparing Henoch-Schonlein purpura nephritis with IgA nephropathy in children].

OBJECTIVE: Henoch-Schonlein purpura nephritis (HSPN) and IgA nephropathy are very similar in immunopathological changes, and therefore some nephrologists considered that they are substantially one disease entity caused by IgA immune abnormalities, and IgA nephropathy is, in fact, a kind of HSPN without rashes. The present study aimed to characterize their relationship through clinico-pathological comparison between IgA nephropathy and HSPN. METHODS: Thirty-one children with IgA nephropathy aged from 3 to 15 years and 120 children with HSPN aged between 4 to 15 years were enrolled in this study. Their clinical manifestations, blood biochemical test, serum immunology and follow-up data were collected and analyzed. Renal pathological findings in light microscopy, immunofluorescence and electron microscopy were analyzed and also compared between 31 children with IgA nephropathy and 32 children with HSPN. RESULTS: The age of onset was over 12 years in 25.8% children with IgA nephropathy, but only in 10% with HSPN, and the difference was significant (P < 0.05). The clinical patterns of IgA nephropathy and HSPN were similar, but extra-renal manifestations were seen more often in HSPN, all of them had skin purpura, 59% had gastrointestinal symptoms and 47% suffered from arthralgia. While the abdominal pain occurred only in 3.2% children with IgA nephropathy. The renal pathological investigation showed global sclerosis in 35.5% of IgA nephropathy and 3.1% of HSPN, mesangial sclerosis in 41.9% of IgA nephropathy and 6.3% of HSPN, but endothelial proliferation in 65.6% of HSPN and 29% of IgA nephropathy. The differences were extremely significant (P < 0.01). Thin basement membrane nephropathy were only found in 6.5% children with IgA nephropathy, but in none with HSPN. The electronic dense deposits in HSPN were sparse, loose and widely spread in glomerular mesangium, subendothelial area and even intra basement membrane. While the deposits were dense, lumpy and mostly limited in mesangium and paramesangium in IgA nephropathy. IgG was found in glomerular immune deposits in 71.9% of HSPN, but only 19.4% of IgA nephropathy. No IgG deposit was observed in 81.6% of IgA nephropathy, among them most showed IgA and IgM and/or C(3) deposit. Predominant IgG deposits were found in 12.5% of HSPN with relatively weak IgA deposit, moreover 6.3% of HSPN showed linear IgG deposits in glomerular capillary wall, which couldn't be found in IgA nephropathy. The follow-up data of average 20 months showed complete remission in 72.5% of HSPN and 19.4% in IgA nephropathy after 34 months follow-up. Moreover, 64.5% of IgA nephropathy had consistent hematuria and proteinuria, 16.1% had active nephritides, the difference was significant (P < 0.05). CONCLUSION: Significant clinico-pathological differences were found between HSPN and IgA nephropathy, which does not support the one disease entity hypothesis. HSPN and IgA nephropathy are probably two diseases with similar immune abnormalities.

Adolescent↗

PubMed2002

A simple random amplified polymorphic DNA genotyping method for field isolates of Dermatophilus congolensis.

Dermatophilus congolensis is the pathogenic actinomycete that causes dermatophilosis in cattle, lumpy wool in sheep and rain scald in horses. Phenotypic variation between isolates has previously been described, but its genetic basis, extent and importance have not been investigated. Standard DNA extraction methods are not always successful for D. congolensis due to its complex life cycle, one stage of which is encapsulated. Here we describe the development of rapid and reliable DNA extraction and random amplified polymorphic DNA (RAPD) methods that can be used for genotyping D. congolensis field isolates. Our results suggest that genotypic variation between isolates correlates with host species. Several DNA extraction methods and RAPD protocols were compared. An extraction method based on incubation of the bacterium in lysozyme, sodium dodecyl sulphate (SDS) and proteinase K treatments and phenolic extraction yielded high-quality DNA, which was used to optimize RAPD-polymerase chain reaction (PCR) protocols for two random primers. An alternative rapid, non-phenolic extraction method based on proteinase K treatment and thermal shock was selected for routine RAPD typing of isolates. DNA extracted from reference strains from cattle, sheep and horse using either method gave reproducible banding patterns with different DNA batches and different thermal cyclers. The rapid DNA extraction method and RAPD-PCR were applied to 38 D. congolensis field isolates. The band patterns of the field and type isolates correlated with host species but not with geographical location.

Actinomycetales↗

PubMed1997

Electrophoretic and antigenic characterisation of Dermatophilus congolensis extracellular products.

Dermatophilus congolensis is the causative agent of bovine dermatophilosis and lumpy wool in sheep. Two field isolates of D. congolensis, one each from a cow in Ghana and a sheep in Scotland, were cultured for 24-72 h in a synthetic medium based on RPMI-1640. Culture filtrates were examined by SDS-PAGE and considered to contain extracellular products released by growing hyphae and filaments. Electrophoretic profiles of culture filtrates of the two isolates contained common bands and bands that were unique to each isolate. The composition of extracellular products altered with increasing culture periods indicating that specific products were released at different stages of growth. Culture filtrate prepared in the presence of serine protease and metalloprotease inhibitors contained more and better defined bands than that prepared without protease inhibitors indicating the presence of proteases in culture filtrates. Western blot analysis of extracellular products using a panel of sera showed that the two isolates from different host species and distant geographical locations contained cross-reactive antigens. Natural and experimental infections stimulated antibody responses to antigens in culture filtrates, sera from animals that were disease free but in-contact with dermatophilosis-infected animals also contained antibodies to extracellular antigens. The antigens recognised by most sera had molecular weights of 200 kDa in the bovine isolate, 170 kDa in the ovine isolate and 67, 27 and 52-55 kDa in both isolates. The number of antigenic bands of both isolates was positively correlated with the intensity of challenge and the severity of infection: antibodies in sera from disease-free cattle in Ghana recognised more antigens than sera from disease-free sheep in Scotland and more antigens were recognised by sera from chronically-infected Ghanaian cattle than by sera from experimentally-infected calves and sheep. The latter developed antibodies to antigens of 27 and 24 kDa during the course of infection. The electrophoretic profiles of extracellular products of D. congolensis are less complex than those of other structures of the bacterium yet they exhibit differences between the two isolates. Extracellular products contain antigens recognised by sera from naturally exposed and experimentally-infected animals that may be involved in immunity to D. congolensis or immunopathogenesis of dermatophilosis.

Actinomycetales↗