l-tryptophan-related eosinophilia-myalgia syndrome possibly associated with a chronic B-lymphocytic leukemia

Böhme, Angelika; Wolter, M.; Hoelzer, Dieter

doi:10.1007/s002770050449

Cited by 4 publications

(3 citation statements)

References 34 publications

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“…Of greatest potential concern is the development of hematologic abnormalities and myelodysplastic syndromes. Aplastic anemia, hemolytic anemia, thrombocytopenia, lymphoma, and B lymphocytic leukemia have been described in patients with EF [24,[28][29][30][31]. The pathogenesis of the hematologic complications of EF is unknown.…”

Section: Eosinophilic Fasciitismentioning

confidence: 99%

Scleroderma-like cutaneous syndromes

Mori¹,

Kahari²,

Varga

2002

Curr Rheumatol Rep

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Several distinct entities associated with dermal fibrosis can mimic scleroderma/systemic sclerosis. The list of scleroderma-like conditions or scleroderma variants includes eosinophilic fasciitis, localized forms of scleroderma, scleredema and scleromyxedema, keloids, and environmental exposure-associated conditions including eosinophilia-myalgia syndrome and pseudosclerodermas induced by various drugs. Although these conditions are relatively uncommon, their accurate recognition is essential to avoid misdiagnosis and inappropriate therapy. The pathogenesis of these scleroderma variants appears to share similarities with each other and with that of scleroderma. Better understanding of scleroderma-like disorders is emerging through epidemiologic investigations, and in vivo and in vitro experimental research. Activation of eosinophils and disordered regulation of fibroblast collagen synthesis, apoptosis, and proliferation are recurrent findings in these disorders. The etiologic role of infection with Borrelia species or other microorganisms remains controversial. Cytokines such as transforming growth factor-beta, interleukin-4, interleukin-13, and connective tissue growth factor contribute to fibrosis in these disorders by inducing an accentuated and persistent fibrogenic response to tissue injury. The role of genetic factors in susceptibility and clinical expression of scleroderma-like conditions remains to be systematically addressed. Because of the relative rarity of these conditions, few well-controlled clinical treatment trials have been performed. In addition, there is no consensus on optimal management. Much anecdotal information and small clinical series indicate that phototherapy may have a role in the treatment of scleroderma-like conditions.

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Section: Eosinophilic Fasciitismentioning

confidence: 99%

Scleroderma-like cutaneous syndromes

Mori¹,

Kahari²,

Varga

2002

Curr Rheumatol Rep

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“…In rare cases, HES has been associated with the development of hematological malignancies. 1, 11 Neuromuscular manifestations of hypereosinophilia occur with parasitic infection (trichinosis), after toxin exposure, as a manifestation of HES, or as an isolated eosinophilic neuromuscular disorder. Eosinophilic syndromes limited to muscle have a better prognosis than generalized HES.…”

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confidence: 99%

Chronic eosinophilic perimyositis with persistent myalgias

Živković¹,

Lacomis²,

Clemens³

2002

Muscle and Nerve

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A 43-year-old man with an 8-year history of hypereosinophilia was evaluated for persistent muscle pain. Methotrexate and corticosteroids were ineffective. Examination, limited by pain even with passive motion, showed only mild weakness. Electromyography and muscle enzymes were normal. A needle muscle biopsy specimen revealed eosinophilic perimyositis. This case illustrates that the diagnosis of eosinophilic perimyositis requires histopathological evaluation, which should be pursued in patients with eosinophilia and persistent myalgia despite normal electromyography and muscle enzymes.

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“…Most of these autoimmune phenomena also have been associated with mycoplasma infections in humans (15,37). While the etiology of EF has remained obscure, the following remarks should be noted: (i) the presence of granzyme B-positive CD8 ϩ T lymphocytes observed in the EF skin lesions suggests a cytotoxic immune response to infectious or environmental agents (34); (ii) levotryptophan (LT) consumption, presumably containing biological or chemical impurities, has been associated with EF or eosinophilia-myalgia syndrome (EMS) (2,(8)(9)(10)(11)(12). Here we report a young EF patient with mycoplasma bacteremia (M. arginini) and possibly concurrent infection, or previously infected-recently recovered status, with another mycoplasma species (M. pneumoniae), who was successfully treated with high-dose corticosteroids in combination with long-term high doses of antibiotics.…”

mentioning

confidence: 99%

Eosinophilic Fasciitis Associated with Mycoplasma arginini Infection

et al. 2012

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Eosinophilic fasciitis (EF) with generalized sclerodermiform skin lesions developed over a 19-month period in a previously healthy 23-year-old man. Although we confirmed EF by skin histology and laboratory tests, the recurrent fevers and the clinical observation of sclerotic prepuce with urethritis indicated further bacteriological analysis by conventional microbiological and DNA-based tests. Urethra cultures were positive for an arginine-hydrolyzing mycoplasma and Ureaplasma urealyticum. The patient also had serum IgM antibodies to Mycoplasma pneumoniae using enzyme-linked immunosorbent assay (ELISA)-based qualitative detection. Mycoplasma arginini was isolated from two independent venous blood serum samples and was identified by conventional microbiological tests and sequencing of the 16S rRNA and rpoB genes (GenBank sequence accession numbers HM179555 and HM179556, respectively). M. arginini genomic DNA also was detected by species-specific PCR in the skin lesion biopsy sample. Treatment with corticosteroids and long-term courses of selected antibiotics led to remission of skin symptoms and normalization of laboratory values. This report provides the first evidence of EF associated with mycoplasma infection and the second report of human infection with M. arginini and therefore suggests that this mycoplasma infection might have contributed to the pathogenesis of the disease. CASE REPORTA 23-year-old male, a former bodybuilder, sought medical treatment for progressive generalized skin tightening. His previous medical history was unremarkable. The patient had terminated sport exercises 19 months earlier due to a sudden, progressive skin induration, which led to restricted movement of wrists and ankles within several months, and cutaneous symptoms gradually extended to the extremities and to the upper body. At the beginning of his illness, he quit body building and stopped anabolic steroid consumption. Within 1 year, he lost 23 kg, he became febrile in the afternoons, and his skin disease slowly progressed. Serological tests for HIV, hepatitis C virus (HCV), and Lyme borreliosis were negative. His crista biopsy specimen revealed eosinophilia. The patient was treated with a very low dose of prednisone (10 mg/day orally [p.o.]) for the next 6 months without dermatological improvement. Any attempt to decrease the prednisone dosage resulted in return of arthralgia and fever.Upon initial examination, the patient suffered from generalized skin tightening and tautness on his whole trunk and on all the extremities except the fingers and toes. He also exhibited multiple cutaneous venous furrowing on the arms (Fig. 1). The shiny, brownish, firm skin with some porcelain-whitish patches was tightly bound to the underlying tissues, resulting in a restricted movement of the chest, wrists, elbows, ankles, and knees. The genital region was also involved; the indurated, sclerotic, depigmented prepuce led to phimosis and showed lichen sclerosus-like symptoms with an inflamed urethral orifice.Initial laboratory tests showed periph...

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l-tryptophan-related eosinophilia-myalgia syndrome possibly associated with a chronic B-lymphocytic leukemia

Cited by 4 publications

References 34 publications

Scleroderma-like cutaneous syndromes

Scleroderma-like cutaneous syndromes

Chronic eosinophilic perimyositis with persistent myalgias

Eosinophilic Fasciitis Associated with Mycoplasma arginini Infection

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