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PubMed2004

Skin disorders with prominent eosinophilic infiltration treated successfully with nicotine. Report of two cases.

BACKGROUND: Nicotine was effective in the treatment of skin disorders attributable to neutrophils such as pyoderma gangrenosum and orogenital ulcerations due to Behçet's disease. OBJECTIVE: We assessed the efficacy of nicotine for treating skin disorders with prominent eosinophilic infiltration. PATIENTS AND METHODS: A 67-year-old man with Kimura's disease was treated for 2 weeks with nicotine chewing gum (6 mg/day). A 50-year-old woman with erythema nodosum with eosinophilic infiltration was treated for 4 weeks with transdermal nicotine patches (5 mg/day). RESULTS: Both patients showed a dramatic clinical and histopathological improvement. CONCLUSION: Our results suggest that nicotine may be useful for treating skin disorders with eosinophilic infiltration as well as neutrophilic dermatoses.

Administration, Topical↗
PubMed1990

Cutaneous manifestations of Takayasu arteritis. A retrospective study of 80 cases.

The aim of this retrospective study is to delineate in Europe the frequency and type of cutaneous manifestations associated with Takayasu arteritis (TA). Eighty patients with TA were analyzed. Symptoms suggestive of Raynaud's syndrome were noted in 11 patients (14%) and could be directly related to large vessel involvement. Other skin lesions were observed in 10 patients (12.5%). Five had acute tender erythematous nodules on the legs with a clinical diagnosis of erythema nodosum; 2 had subacute ulcerated nodules of the legs; 1 had pyoderma-gangrenosum-like ulcerations of the four limbs which resulted from the breakdown of subcutaneous nodules; 1 had lupus-like malar flush, and the last one had urticarial lesions with livedo reticularis. Skin samples were obtained from 4 patients. Three of them agreed that reiterated biopsies be done on recurrent lesions. A granulomatous vasculitis was observed in 2 cases involving hypodermal arterioles in one case and veins in the other. The other pathological findings were septal and lobular panniculitis which can be associated with granulomatous vasculitis. Different histological findings on reiterated biopsies were frequently found. The absence of any other etiology and chronological arguments suggested a relationship between these skin lesions and TA. Tuberculosis was probable in 1 case but apparently was not related to the skin lesions.

Adult↗
PubMed1975

A case of cutaneous amoebiasis.

The present report concerns a patient with ulceration around an artificial anus. The condition had been treated for some time as pyoderma gangrenosum but finally proved to be cutaneous amoebiasis; the presence of Entamoeba histolytica was demonstrated. A cure was achieved with metronidazol (Flagy) and diloxanide furoate (Furamide) given orally.

Acetanilides↗
PubMedDate not supplied

Inflammatory diseases associated with Takayasu's arteritis.

The etiology of Takayasu's arteritis is unknown; however, abnormality of the immune system plays an important role. To clarify a possible association between inflammatory diseases and Takayasu's arteritis, the authors retrospectively reviewed the medical records of 36 patients (2 men and 34 women), admitted to their department with this diagnosis. They found that 11 of the 36 patients had at least 1 other chronic or subacute inflammatory disease: Chronic tonsillitis, tuberculous lymphadenitis of the neck, chronic hepatitis B (infectious diseases); pyoderma gangrenosum, erythema nodosum (skin lesions); subacute thyroiditis, chronic thyroiditis (autoimmune diseases); Crohn disease, and ulcerative colitis (inflammatory diseases with unknown etiology). The present observation revealed a high prevalence of other inflammatory diseases in patients with Takayasu's arteritis and raises the possibility that Takayasu's arteritis may be associated with abnormality of the immune system activated by other inflammatory diseases, such as infection, autoimmune diseases, or inflammatory diseases of unknown origin.

Adolescent↗
PubMedDate not supplied

Topical cyclosporin in the treatment of dermatologic diseases.

Cyclosporine A (CsA) has been in clinical use for some decades, primarily for the prevention and treatment of organ transplant rejection and graft-versus-host disease. In more recent years, Cyclosporine has been recognized as beneficial in the treatment of dermatologic diseases, such as: psoriasis, lichen planus, Behcet disease, atopic dermatitis, pyoderma gangrenosum and epidermolysis bullosa acquisita. Above all, Cyclosporine is an important therapeutic modality for several dermatologic diseases that are refractory to other agents.

Journal Article↗
PubMed2006

Juvenile systemic lupus erythematosus in multicase families from Saudi Arabia: comparison of clinical and laboratory variables with sporadic cases.

The object of this study was to compare patients with familial versus sporadic systemic lupus erythematosus (SLE) with respect to clinical, laboratory variables and outcome. The familial SLE group comprised 12 patients while the comparative group comprised 24 patients selected by systemic sampling from our pediatric rheumatology clinic database. Those patients are listed according to the date of referral, which represents a sampling frame. The first patient was chosen randomly and subsequent patients were chosen at intervals of three. The two groups were compared with respect to: demographic information, age of onset of SLE, disease and follow up duration, clinical and laboratory variables and outcome. The patients from the familial group were younger and had an earlier age of onset of disease (P = 0.03, 0.001 respectively). Seven patients with familial SLE were from the eastern region of Saudi Arabia (P = 0.006). The two groups were comparable with respect to gender, disease duration and follow-up. At diagnosis, the discoid rash was more frequent in the familial group (P = 0.03) while other clinical and laboratory variables including disease activity as measured by SLEDAI did not show significant differences. The mean dose of steroid and use of other immunosuppressive therapy were similar in both groups. Three patients from the familial group died; two of them had unusual complications (one patient had transverse myelitis and pancreatic pseudocyst and the other one had extensive pyoderma gangrenosum). All patients from the sporadic group are alive in stable condition but one patient had severe central nervous system disease. Familial SLE patients tend to be younger and more likely to have discoid rash, in addition a marked difference in the origin of patients was noted. These differences may be helpful in identifying SLE patients with a stronger genetic predisposition. The mortality among familial SLE patients is more frequent which may reflect the disease severity.

Adolescent↗
PubMed2003

Leg ulcers of unusual causes.

In this review, unusual causes of leg ulcers are examined with an emphasis on pathophysiology, clinical presentation, and epidemiology. Cutaneous ulcers due to malignancy of unusual leg ulcers with hematologic disorders, vasculitis, sarcoidosis, calciphylaxis, Buerger's disease, and pyoderma gangrenosum are discussed.

Journal Article↗
PubMed2002

Thalidomide-induced neuropathy.

BACKGROUND: Thalidomide is effective for the treatment of some refractory dermatologic and oncologic diseases. Toxic neuropathy limits its use, as embryopathy can be avoided by contraceptive measures. OBJECTIVE: To describe the clinical, electrophysiologic, and pathologic features of thalidomide-induced peripheral neuropathy. METHODS: Clinical and electrophysiologic examinations were performed in seven patients with thalidomide-induced peripheral neuropathy. Thalidomide was used for graft-vs-host disease, pyoderma gangrenosum, and discoid lupus with dosages ranging from 100 to 1,200 mg/day for 5 to 16 months (cumulative dosages of 24 to 384 g). RESULTS: All seven patients had clinical and electrophysiologic evidence of a sensory more than motor, axonal, length-dependent polyneuropathy that presented as painful paresthesias or numbness. Sural nerve biopsies, done in three patients, showed evidence of Wallerian degeneration and loss of myelinated fibers. The symptoms, signs, and electrophysiologic data correlated with total cumulative dose of thalidomide. CONCLUSIONS: Thalidomide induces a dose-dependent sensorimotor length-dependent axonal neuropathy; it should be judiciously used with close neurologic monitoring.

Adult↗
PubMed1988

Trigeminal trophic syndrome.

Trigeminal trophic syndrome is an unusual condition also known as trigeminal neurotrophic ulceration or trigeminal neuropathy with nasal ulceration. The diagnosis is suggested when ulceration of the face, especially of the ala nasi, occurs in a dermatome of the trigeminal nerve that has been rendered anesthetic by a surgical or other process involving the trigeminal nerve or its central sensory connections. A history of paresthesias and self-induced trauma to the area further support the diagnosis. Neurological deficits causing trigeminal trophic syndrome may result from surgical trigeminal ablation, vascular disorders and infarction of the brainstem, acoustic neuroma, postencephalitic parkinsonism, and syringobulbia. The following etiologies of nasal ulceration should be excluded: postsurgical herpetic reactivation and ulceration, syphilis, leishmaniasis, leprous trigeminal neuritis, yaws, blastomycosis, paracoccidioidomycosis, lethal midline granuloma, pyoderma gangrenosum, Wegener's granulomatosis, and basal cell carcinoma. In the case reported here, the diagnosis of TTS was made primarily as a result of previous experience with the syndrome, underscoring the importance of physician recognition of this unusual disorder.

Cerebrovascular Disorders↗
PubMed1993

Cyclosporin. A review of its pharmacodynamic and pharmacokinetic properties, and therapeutic use in immunoregulatory disorders.

Cyclosporin is a lipophilic cyclic polypeptide which produces calcium-dependent, specific, reversible inhibition of transcription of interleukin-2 and several other cytokines, most notably in T helper lymphocytes. This reduces the production of a range of cytokines, inhibiting the activation and/or maturation of various cell types, including those involved in cell-mediated immunity. Thus, cyclosporin has immunosuppressive properties, and has a proven place as first line therapy in the prophylaxis and treatment of transplant rejection. Cyclosporin has also been evaluated in a large range of disorders where immunoregulatory dysfunction is a suspected or proven aetiological factor, and this is the focus of the present review. In patients with severe disease refractory to standard treatment, oral cyclosporin is an effective therapy in acute ocular Behçet's syndrome, endogenous uveitis, psoriasis, atopic dermatitis, rheumatoid arthritis, active Crohn's disease and nephrotic syndrome. Concomitant low dose corticosteroid therapy may improve response rates in some disorders. The drug can be considered as a first line therapy in patients with moderate or severe aplastic anaemia who are ineligible for bone marrow transplantation, with the additional benefit of reducing platelet alloantibody titres. It may also be of considerable therapeutic benefit in patients with primary biliary cirrhosis, particularly those with less advanced disease. Limited evidence suggests cyclosporin is effective in patients with intractable pyoderma gangrenosum, polymyositis/dermatomyositis or severe, corticosteroid-dependent asthma. Indeed, the steroid-sparing effect of cyclosporin is a significant advantage in a number of indications. Furthermore, the drug has shown some efficacy in a wide range of other, generally uncommon disorders in which controlled clinical trials are lacking and/or are unlikely to be performed. Cyclosporin does not appear to be effective in patients with allergic contact dermatitis, multiple sclerosis or amyotrophic lateral sclerosis. It is only temporarily effective in patients with type I (insulin-dependent) diabetes mellitus and should not be used in this indication. To avoid relapse after control of active disease, patients should receive cyclosporin maintenance therapy at the lowest effective dosage. However, maintenance therapy appears to be of no benefit in patients with Crohn's disease and cyclosporin should be discontinued in these patients once active disease is controlled. Hypertrichosis, gingival hyperplasia, and neurological and gastrointestinal effects are the most common adverse events in cyclosporin recipients, but are usually mild to moderate and resolve on dosage reduction. Changes in laboratory variables indicating renal dysfunction are relatively common, although serious irreversible damage is rare.(ABSTRACT TRUNCATED AT 400 WORDS)

Arthritis, Rheumatoid↗
PubMed2000

Potential novel uses of thalidomide: focus on palliative care.

Thalidomide, after being banned from the market in the early 1960s because of the worldwide teratogenesis disaster, is currently being rediscovered because of its multiple therapeutic effects in various serious diseases and symptoms. Original studies examined the anxiolytic, mild hypnotic, anti-emetic and adjuvant analgesic properties of this drug. Subsequently, thalidomide was found to be highly effective in managing the cutaneous manifestations of leprosy (erythema nodosum leprosum) and even to be superior to aspirin (acetylsalicylic acid) in controlling leprosy-associated fever. Recent research shows promising results with thalidomide in patients with progressive bodyweight loss related to advanced cancer and HIV infection. Thalidomide therapy of diseases such as tuberculosis, sarcoidosis, aphthous ulcers in HIV syndrome and Behcet's disease, rheumatoid arthritis, multiple myeloma, graft-versus-host disease, pyoderma gangrenosum, inflammatory bowel disease, Sjögren's syndrome, lupus erythematosus and a variety of solid tumours is currently being explored. Furthermore, in preliminary studies, thalidomide has been found to be effective in several syndromes related to advanced cancer, such as the cancer cachexia syndrome, chronic nausea, insomnia, profuse sweating and pain. Whether thalidomide has a therapeutic effect on neoplastic fever has yet to be elucidated. These intriguing features make the use of the drug potentially attractive for palliative care. In addition, by a distinct mechanism of action compared with most other drugs, thalidomide offers the possibility of combined treatment with other agents with non-overlapping toxicities. The mechanism of action of thalidomide is probably based on the suppression of tumour necrosis factor-alpha and the modulation of interleukins. However, it is not possible to identify a single dominant mechanism, since the action of cytokines and the effect of thalidomide appear to be complex. This review article discusses the original uses and teratogenic effects of thalidomide within its historical context and, linking recent research at the molecular level with clinical findings, aims to provide the reader with insight into the current understanding of its biological actions, toxicities and potential benefits.

Angiogenesis Inhibitors↗
PubMed2005

Molecular and genetic characteristics of hereditary autoinflammatory diseases.

Autoinflammatory diseases are defined as recurrent "unprovoked" inflammatory events which do not produce high-titer autoantibodies or antigen-specific T cells. There are currently eight hereditary forms of these diseases: Familial Mediterranean fever (FMF), hyperimmunoglobulinemia D with periodic fever syndrome (HIDS), tumor necrosis factor receptor-associated periodic syndrome (TRAPS), Muckle-Wells syndrome (MWS), familial cold autoinflammatory syndrome (FCAS), chronic infantile neurologic cutaneous articular (CINCA) syndrome or neonatal-onset multisystem inflammatory disease (NOMID), pyogenic sterile arthritis, pyoderma gangrenosum, acne (PAPA) and Blau syndrome. Apart from FMF (which has a prevalence of about 0.1 percent among non-Ashkenazi Jews, Armenians, Turks and Arabs), they are very rare disorders. FMF and HIDS are autosomal recessive diseases, all the other members of the family are autosomal and dominantly transmitted. Their common clinical features are recurrent and usually short attacks of synovitis and various skin eruptions; abdominal pain and fever are also frequently observed. The genes of all of these diseases have been discovered and, with the exception of HIDS, it was found that the proteins they encode share certain domains taking part in innate immunity and apoptosis. Thus it was evident that hereditary autoinflammatory diseases may help us understand better a number of important and prevalent pathologic events. We have reviewed the recent and rapidly accumulating knowledge on the molecular aspects of these disorders.

Familial Mediterranean Fever↗
PubMed2001

Evaluation of children with myelodysplastic syndrome: importance of extramedullary disease as a presenting symptom.

Thirty-three children diagnosed with primary myelodysplastic syndrome (MDS) in a single institution over an 8 year period were evaluated with special emphasis on children who presented with extramedullary disease (EMD). EMD was present at diagnosis in 12 (36%) of the 33 children with MDS. Three patients with juvenile myelomonocytic leukemia (JMML) and 2 patients with chronic myelomonocytic leukemia (CMML) presented with pleural effusion. Pericardial effusion was present in 3 of these patients, two of whom also had thrombosis. Pyoderma gangrenosum, relapsing polychondritis were the initial findings in another two cases with JMML. Lymphadenopathy (n=1), gingival hypertrophy (n=2), orbital granulocytic sarcoma (n=1) and spinal mass (n=1) were the presenting findings in 5 patients with refractory anemia with excess of blasts in transformation. Since high-dose methylprednisolone (HDMP, 20-30 mg/kg/day) has been shown to induce differentiation and apoptosis of myeloid leukemic cells in children with different morphological subtypes of acute myeloid leukemia in vivo and in vitro, 25 children with de novo MDS were treated with combined HDMP and cytotoxic chemotherapy. Dramatic improvement of EMD and decrease in blast cells both in the peripheral blood and bone marrow were obtained following administration of short-course HDMP treatment alone as observed in children with AML. HDMP, combined with low-dose cytosine arabinoside and mitoxantrone were used for the remission induction. Remission was achieved in 8 (80%) of 10 children who presented with EMD and in 9 (60%) of 15 children without EMD. Long-term remission (>6 years) was obtained in 4 (two with JMML and two with CMML), three of whom presented with EMD. In conclusion EMD can be a presenting finding in childhood MDS as observed in adults. In addition, the beneficial effect of HDMP combined with more intensive chemotherapy should be explored as alternative therapy in children with MDS not suitable for bone marrow transplantation.

Adolescent↗
PubMed1997

Dermatologic manifestation of Behçet's disease.

Dermatologic lesions in Behçet's disease are regarded as important manifestation for diagnosis. Various kinds of cutaneous lesions appear in patients with Behçet's disease. They present as erythema nodosum-like lesion, papulopustular eruption, erythema multiforme-like lesion, thrombophlebitis, skin ulcer, Sweet's syndrome-like lesion, bullous necrotizing vasculitis, and pyoderma gangrenosum. The hyperreactivity of the skin to any intracutaneous injection or needle prick, which is known as pathergy, is one clinically-unique feature of the disease.

Behcet Syndrome↗
PubMed2005

A case of erythema nodosum and serositis associated with myelodysplastic syndrome.

Myelodysplastic syndrome (MDS) is a heterogenous group of stem cell disorders usually characterized by progressive refractory cytopenias, which could progress to acute myeloid leukemia. MDS may be associated with a wide spectrum of skin lesions, including neoplastic cell infiltration, Sweet's syndrome, pyoderma gangrenosum, erythema elevatum diutinum, vasculitis, and panniculitis. However, erythema nodosum is rarely associated with MDS. Unusual rheumatologic manifestations in patients with MDS also have been reported, which range from asymptomatic serological abnormalities to classic connective tissue disorders such as Sjögren's syndrome, relapsing polychondritis, systemic lupus erythematosus, rheumatoid arthritis and mixed connective tissue disease. However, concurrent erythema nodosum and serositis has rarely been reported. We describe a case of MDS with erythema nodosum and immune-mediated pericardial effusion in a 34-year-old woman.

Adult↗
PubMed1996

Cyclosporine in the treatment of dermatologic disease: an update.

Treatment with cyclosporine is beneficial for many dermatologic diseases such as psoriasis, lichen planus, Behçet disease, atopic dermatitis, pyoderma gangrenosum, and epidermolysis bullosa acquisita. The selective action of cyclosporine on helper T cells and its rapid therapeutic action and weak myelotoxicity are the key advantages in the treatment of many dermatologic diseases. Nevertheless, drug toxicity, especially nephrotoxicity, high rates of relapse after treatment cessation, and high cost have limited its use to those diseases refractory to other therapies. Herein we discuss the use of cyclosporine for dermatologic diseases relative to efficacy, dosage, safety profile, and monitoring. In addition, we review the formulations and metabolism of cyclosporine; discuss its mechanism of action, clinical indications in dermatology, and side effects; and provide usage guidelines for this drug. Cyclosporine can be safely administered when potential toxicities, dosing, and monitoring guidelines are known.

Cyclosporine↗
PubMed1990

Leg ulcers in rheumatoid arthritis.

A retrospective study over an eight-year period of 33 episodes of leg ulceration in 26 patients with rheumatoid arthritis requiring inpatient management is reported. the aetiology of the ulcers was found to be multifactorial. The most common factors were venous insufficiency (45.5%), trauma or pressure (45.5%) and arterial insufficiency (36.4%). Vasculitis (18.2%) and Felty's syndrome (12.1%) were less frequent causes, and pyoderma gangrenosum was rare. Most patients had seropositive erosive disease with high rheumatoid factor titres and significant functional impairment; over half were on maintenance corticosteroids. Colonisation of the ulcers by organisms, predominantly Staphylococcus aureus, was common (69.7%). Skin grafting was required in 63.3%, but the rate of complete take was only 42.9% despite multiple attempts. Hospitalisation was prolonged (mean 47.9 days) and the recurrence rate requiring further hospitalisation was 26.9%. The diagnosis of vasculitis and the limited role of biopsy in establishing its presence are discussed.

Adult↗
PubMed1982

Spondyloarthropathy associated with hidradenitis suppurative and acne conglobata.

We evaluated 10 patients with hidradenitis suppurative or acne conglobata who developed arthritis. In contrast to patients with acne fulminans and arthritis, all our subjects were adults over 22 years of age; nine were black; and four were women. Nine patients had episodic inflammatory oligoarthritis affecting mainly larger joints of the upper and lower extremities. Eight patients had roentgenographic evidence of peripheral arthritis, four with erosions. Nine had clinical axial arthropathy, but roentgenograms showed abnormalities of the axial skeleton in all 10 patients. Pyoderma gangrenosum, erythema nodosum, conjunctivitis, urethritis, and oral and penile ulcers occurred in some patients. Rheumatoid factor was negative in all patients; the erythrocyte sedimentation rate was elevated in nine; five had chronic anemia; four had circulating immune complexes; and complement components were elevated in four. There was no increased incidence of HLA-B27 or other HLA-B7 cross-reacting antigens. A temporal relation of skin and joint disease activity was suggested. We report a spondyloarthropathy associated with hidradenitis suppurativa and acne conglobata. Clinical and laboratory manifestations suggest the arthropathy may be reactive to chronic cutaneous infection.

Acne Vulgaris↗