Cervical spondylitis and spinal abscess due to Actinomyces meyeri

Human actinomycosis with involvement of the spine is a rare condition although it has been first described a long time ago. It is probably underrecognized since its clinical presentation is often misleading and accurate bacteriological diagnosis is challenging. We herein report a rare case of cervical actinomycosis with paravertebral abscess and spondylitis imputed to an infection by Actinomyces meyeri in a 52-year-old immunocompetent Caucasian man. A. meyeri should be considered as a potential cause for subacute or chronic spondylitis, even in immunocompetent subjects. Modern diagnostic tools such as Matrix-Assisted Laser Desorption–Ionization Time of Flight mass spectrometry and 16S rRNA sequencing are efficient for accurate microbiological identification.

Cervical spondylitis and spinal abscess due to Actinomyces meyeri.

Human actinomycosis with involvement of the spine is a rare condition although it has been first described a long time ago. It is probably underrecognized since its clinical presentation is often misleading and accurate bacteriological diagnosis is challenging. We herein report a rare case of cervical actinomycosis with paravertebral abscess and spondylitis imputed to an infection by Actinomyces meyeri in a 52-year-old immunocompetent Caucasian man. A. meyeri should be considered as a potential cause for subacute or chronic spondylitis, even in immunocompetent subjects. Modern diagnostic tools such as Matrix-Assisted Laser Desorption-Ionization Time of Flight mass spectrometry and 16S rRNA sequencing are efficient for accurate microbiological identification.

Pulmonary streptomyces infection in patient with sarcoidosis, France, 2012.

TO THE EDITOR: Streptomyces spp. are aerobic, gram-positive bacteria of the order Actinomycetales, known for their ability to produce antimicrobial molecules such as streptomycin. Streptomyces spp., usually saprophytic to humans, can cause local cutaneous fistulized nodules known as actinomycetoma or mycetoma. Severe invasive infections have seldom been reported, but most cases reported have occurred in immunocompromised patients (1-5). We report a case of invasive pulmonary infection caused by a Streptomyces sp. in a splenectomized patient with sarcoidosis.

IgG4-related systemic disease: features and treatment response in a French cohort: results of a multicenter registry.

IgG4-related systemic disease is now recognized as a systemic disease that may affect various organs. The diagnosis is usually made in patients who present with elevated IgG4 in serum and tissue infiltration of diseased organs by numerous IgG4+ plasma cells, in the absence of validated diagnosis criteria. We report the clinical, laboratory, and histologic characteristics of 25 patients from a French nationwide cohort. We also report the treatment outcome and show that despite the efficacy of corticosteroids, a second-line treatment is frequently necessary. The clinical findings in our patients are not different from the results of previous reports from Eastern countries. Our laboratory and histologic findings, however, suggest, at least in some patients, a more broad polyclonal B cell activation than the skewed IgG4 switch previously reported. These observations strongly suggest the implication of a T-cell dependent B-cell polyclonal activation in IgG4-related systemic disease, probably at least in part under the control of T helper follicular cells.

Tocilizumab in refractory adult Still's disease.

OBJECTIVE: There is an unmet need for the treatment of adult Still's disease (ASD), the pathogenesis of which may involve interleukin-6 (IL-6). We report the first series of patients with ASD treated with tocilizumab (TCZ), a humanized anti-IL-6 receptor antibody. METHODS: All ASD patients treated with TCZ in France between July 2006 and July 2009 after failure to all available therapies were included in this cohort study. The main outcome measures were the European League Against Rheumatism (EULAR) improvement criteria and resolution of systemic symptoms at the 3- and 6-month followup periods. RESULTS: Fourteen patients with refractory ASD were included. At the start of TCZ treatment, despite a mean prednisone dosage of 23.3 mg/day, based on a 28-joint count, mean tender joints were 10.5, mean swollen joints were 7.9, and the mean Disease Activity Score in 28 joints was 5.61. Recurrent systemic involvement, including fever and rash, was present in 7 patients. TCZ was administered at 5-8 mg/kg every 2 or 4 weeks (8 mg/kg/month, n = 9). Eleven patients successfully completed the 6-month study; 1 withdrew due to necrotizing angiodermatitis, another due to chest pain at each TCZ infusion, and a third due to systemic flare. A good EULAR response was observed in 64% of patients (9 of 14) at 3 months and EULAR remission was observed in 57% (8 of 14) at 6 months. Systemic symptoms were resolved in 86% of patients (6 of 7). Moreover, corticosteroid dose was reduced by 56%. No other severe adverse effects occurred. CONCLUSION: TCZ is a promising new treatment for ASD.

Assessment of liver fibrosis with transient elastography and FibroTest in patients treated with methotrexate for chronic inflammatory diseases: a case-control study.

BACKGROUND & AIMS: Although methotrexate (MTX) is used in the effective treatment of inflammatory disorders, its use is hampered by the risk of liver fibrosis. Non-invasive methods for the diagnosis of liver fibrosis, such as transient elastography (FibroScan) and FibroTest could be useful for monitoring MTX-liver toxicity. The aim of this case-control study was to determine factors associated with liver fibrosis in a large cohort of patients requiring MTX. METHODS: Consecutive adults with various benign inflammatory diseases were prospectively assessed using FibroScan and FibroTest when they were treated with MTX (cases) or before beginning treatment (controls). RESULTS: Among 518 included patients, 44 patients (8.5%) had FibroScan and/or FibroTest results suggesting severe liver fibrosis. In a multivariate analysis, factors associated with abnormal markers of liver fibrosis were the body mass index >28 kg/m(2) and high alcohol consumption. Neither long MTX duration nor cumulative doses were associated with elevated FibroScan or FibroTest results. CONCLUSIONS: Severe liver fibrosis is a rare event in patients treated with MTX and is probably unrelated to the total dose. Patients with other risk factors for liver disease should be closely monitored with non-invasive methods before and during MTX treatment.

Autochthonous leprosy in metropolitan France presenting with a diffuse infiltration of the face and febrile illness.

Leprosy is now considered in Western Europe as a strictly imported disease. A case of presumed locally acquired diffuse lepromatous leprosy was observed in a native-Portuguese woman living in France who developed an acute febrile presentation with extensive cutaneous signs.

Rituximab Efficacy during a Refractory Polyarteritis Nodosa Flare.

Polyarteritis nodosa (PAN) is a systemic vasculitis whose severe forms are treated with glucocorticoids and cyclophosphamide. Refractory patients are exposed to many complications, notably accelerated atherosclerosis. We report a case report of 71-year-old man followed for polyarteritis nodosa refractory to glucocorticoids and cyclosphosphamide. Systemic vasculitis relapses are followed to accelerated atherosclerosis: severe ischemic lesions led to amputation of lower limbs. Remission of refractory PAN is obtained with rituximab. Disappearance of biological inflammatory is allowed to regression of ischemic lesions in upper limbs. In this situation, we recommend a systematic vascular work-up for patients suffered from refractory vasculitis. On the other hand, therapeutic trials are needed to determine the real efficacy and place of rituximab in the treatment of polyarteritis nodosa.

Extensive cutaneous larva migrans with folliculitis mimicking multimetameric herpes zoster presentation in an adult traveler returning from Thailand.

Hookworm-related cutaneous larva migrans (CLM) is a frequent cutaneous disease among travelers returning from the tropics. It can be misdiagnosed or treated incorrectly. We present a 42-year-old French patient who contracted the disease during a holiday in Thailand and who experienced an extensive CLM syndrome with a less frequent abdominal localization and a pseudo-multimetameric homolateral topography. The condition was late diagnosed and secondarily efficiently cured by a unique administration of ivermectin. Simple anamnestic information--often revealing beach activities--and clinical aspect of the creeping eruption allow to prevent diagnosis delay and to avoid aggressive or inadequate intervention.

[Visceral leishmaniasis in an immunocompetent subject, acquired in the Pyrenees region]. / Leishmaniose viscérale acquise en région pyrénéenne chez un sujet immunocompétent.

INTRODUCTION: Visceral leishmaniasis can develop in immunocompetent subjects exposed to a risk of infestation during physical activities outdoors. CASE: A 35-year-old woman was hospitalized for impaired health status with fever, hepatomegaly, splenomegaly, and pancytopenia, which worsened during her stay. Leishmaniasis was finally diagnosed, based on serologic testing and the finding of Leishmania infantum in the sternal aspiration. Eight months earlier, she had spent two weeks at a lake in the Pyrenees. Treatment with meglumine antimoniate was efficacious. COMMENTS: This case is original because of the extent of the organomegaly and its onset after vacation in an area far from those where it is thought to be endemic.

[Retrospective analysis of 107 imported adult cases of malaria. Experience report of uncomplicated falciparum malaria treatment in adults with oral atovaquone-proguanil]. / Analyse rétrospective de 107 cas de paludisme d'importation chez l'adulte. Intérêt de l'association orale atovaquone-proguanil dans le traitement de l'accès simple à Plasmodium falciparum.

OBJECTIVES: The principal aim of this study was to assess the efficacy of the combination of oral atovaquone and proguanil (AP) in routine treatment of uncomplicated falciparum malaria in recent visitors to the tropics. We also analyzed the epidemiologic, clinical and hematologic characteristics of these patients with imported malaria. METHODS: This retrospective study reviewed the records of 107 cases of imported malaria, treated in the Tropical Disease Unit of Bordeaux University Hospital (France). RESULTS: More than 75% had uncomplicated falciparum malaria; 10 developed complications, and one died. Average time from fever onset to first medical consultation was 4.8 days. Less than 10% had used physical measures of prevention against mosquito bites, and nearly 65% had not used chemoprophylaxis. The most frequent hematologic abnormalities were eosinopenia (84%) and thrombocytopenia (75%). In more than 30% of cases, another disease was also present. The oral AP combination was administered to more than 86% of patients with uncomplicated falciparum malaria (n=72). Tolerance was excellent and only one treatment failure was observed, associated with digestive disorders. CONCLUSION: Better information for travelers and general practitioners is required to reduce the number of cases of imported malaria and to shorten the delay between symptom onset and specific treatment. The oral AP combination appears to be a safe and effective treatment for uncomplicated imported falciparum malaria.

[Celiac disease and autoimmune diseases or systemic disease. Six cases and a review of the literature]. / Maladie coeliaque et maladies auto-immunes ou maladies systémiques.

BACKGROUND: Celiac disease is an autoimmune disorder which may be associated with another autoimmune or systemic disease. OBJECTIVE: To determine the links between autoimmune diseases and celiac disease. PATIENTS AND METHODS: Among 31 patients with a celiac disease, we selected those who had another autoimmune or systemic disease. RESULTS: We report 6 patients with such disease association: 3 with autoimmune thyroiditis including one also with Grave's disease, 2 with systemic lupus erythematosus including one also with insulin-dependent diabetes mellitus, and 1 with temporal arteritis. CONCLUSION: The link between celiac disease and autoimmune thyroiditis or insulin-dependent diabetes mellitus seems to be real but many discrepancies are observed for the other autoimmune diseases. After a literature review, we suggest a summary of effective associations between celiac disease and autoimmune or systemic diseases.

Anti-endothelial cell autoantibodies and soluble markers of endothelial cell dysfunction in systemic lupus erythematosus.

OBJECTIVE: To determine if anti-endothelial cell antibodies (AECA) and plasma markers of endothelial cell function are related to disease severity in systemic lupus erythematosus (SLE). METHODS: We measured AECA by human umbilical vein endothelial cell binding, endothelial markers von Willebrand factor, soluble thrombomodulin, and soluble E-selectin by ELISA, and disease severity by SLEDAI and SLICC/ACR in 35 patients with SLE. RESULTS: Despite high levels of IgG AECA (p = 0.001) and von Willebrand factor (p = 0.0007) compared to 21 healthy controls, we found a positive correlation only between IgG AECA and the SLEDAI index (r = 0.393, p = 0.021). CONCLUSION: IgG AECA seem to be related to disease activity in SLE, possibly in a pathogenic role. Conversely, plasma markers of endothelial cell damage seem to be an epiphenomenon and may simply be related to excess inflammation.

Postvaccinal inflammatory neuropathy: peripheral nerve biopsy in 3 cases.

Autoimmune inflammatory polyneuropathy (PN) can be triggered by vaccination. We report 3 such cases. A 36-year-old female nurse presented 15 days after a hepatitis B vaccination (HBV) with acute sensory disturbances in the lower limbs. She had severe ataxia but no weakness. Cerebrospinal fluid (CSF) protein level was 84 mg/100 mL, with 3 lymphocytes. A 66-year-old man presented 21 days after HBV with severe motor and sensory PN involving all 4 limbs. A 66-year-old man presented 15 days after a yellow fever vaccination with progressive motor and sensory PN involving all 4 limbs and bilateral facial paralysis. CSF protein level was 300 mg/100 mL, with 5 lymphocytes. Six weeks later, a tracheostomy was performed. In these 3 patients, the nerve deficits lasted for months. In each case, peripheral nerve biopsy showed KP1-positive histiocytes but no T-lymphocytes in the endoneurium. On ultrastructural examination, there was axonal degeneration in the first 2 cases; in case 2, a few myelinated fibers exhibited an intra-axonal macrophage but the myelin sheath was preserved. There was only 1 example of macrophage-associated demyelination in case 2, but these were numerous in case 3. It is likely that in the first 2 cases, an autoimmune reaction against some axonal or neuronal components was triggered by HBV. It induced an acute sensory ataxic PN in case 1 and an acute motor and sensory axonal neuropathy (AMSAN) in case 2. The third patient had a chronic inflammatory demyelinating PN, likely triggered by yellow fever vaccination.