[Anterior orbital ischemia secondary to type 1 cryoglobulinemia: A case report and literature review]. / Ischémie du segment antérieur de l'œil secondaire à une cryoglobulinémie de type 1 : à propos d'un cas et revue de la littérature.

INTRODUCTION: Type 1 cryoglobulinemia is characterized by a large number of clinical signs. The lack of specificity of these signs can make diagnosis difficult. Ocular manifestations are rarely described across medical literature. Only 15 cases of ophthalmological involvement secondary to cryoglobulinaemia have been reported. COMMENT: We report the case of a 69-year-old patient with cutaneous type 1 cryoglobulinaemia. He presented with bilateral anterior segment ischemia without retinal involvement with unilateral neovascularisation. Treatment of the B lymphocyte clone with rituximab and bendamustine and plasma exchange were initiated with successfully. Two similar cases describing ischaemic damage to the iris during type 1 cryoglobulinemia have been reported in the literature. CONCLUSION: Irial ischaemia should be considered as a potential in type 1 cryoglobulinaemia.

Elizabethkingia anophelis outbreak in France.

OBJECTIVE: We report an outbreak of Elizabethkingia anophelis infections in France. To the best of our knowledge, this is the first outbreak described in Europe. METHODS: Each E. anophelis-positive microbiological sample was considered a case. All patients were hospitalized in an infectious diseases unit. Clinical, environmental, and microbiological investigations (MALDI-TOF mass spectrometry, PCR, E-test) were performed for each case. RESULTS: Twenty cases were reported from September 2020 to September 2021, mainly community-acquired infections, responsible for nine deaths. The phylogenetic analysis showed a clonal origin and excluded nosocomial transmission. Despite the analysis of multiple environmental specimens, no source of contamination was identified. All strains were highly resistant to cefotaxime, ceftazidime, and imipenem. CONCLUSIONS: Clinicians and microbiologists should be aware of this multidrug-resistant bacterium, capable of causing severe infections. Most strains showed the lowest minimum inhibitory concentration values for cotrimoxazole and ciprofloxacin, making them the best choice for empirical antibiotic therapy.

Telangiectasias of the breasts showing diffuse dermal angiomatosis in a patient with diffuse livedo reticularis.

The breasts are a common location for diffuse dermal angiomatosis (DDA) in a context of obesity and macromastia. The typical clinical presentation includes erythematous or purplish plaques, reticulated telangiectasias, and sometimes livedo reticularis, often complicated by painful ulcerations of the breasts. Biopsy usually confirms a dermal proliferation of endothelial cells staining positively for CD31, CD34 and SMAa and negatively for HHV8. We report herein a woman with DDA of the breasts presenting as diffuse livedo reticularis and acrocyanosis, both long-standing and considered idiopathic following extensive investigations. Since a biopsy of the livedo did not document DDA features in our case, we suggest that our patient's livedo reticularis and telangiectasias could constitute a vascular predisposition for DDA, as its pathogenesis frequently involves an underlying disease involving ischemia, hypoxia, or hypercoagulability.

[Facial ulcerated nodules revealing primary cutaneous CD8-positive aggressive epidermotropic cytotoxic T-cell lymphoma]. / Nodules ulcérés du visage révélant un lymphome T cutané épidermotrope CD8+ cytotoxique agressif.

BACKGROUND: Primary cutaneous CD8+ aggressive, epidermotropic, cytotoxic T-cell lymphoma is a rare disease with a poor prognosis. Herein we report a new case, with facial lesions, which was difficult to diagnose. PATIENTS AND METHODS: A 39-year-old woman was hospitalized for ulcerated nodules on the face that had been developing rapidly for 8 weeks. She had visited Djerba, Tunisia, 3 months earlier. No abnormalities were found on previous routine blood tests. Histopathological analysis of a skin biopsy had revealed non-specific lymphocytic infiltrate. Various therapies, including amoxicillin/clavulanic acid, valaciclovir, corticosteroids, colchicine and doxycycline, proved ineffective. Screening of the cutaneous sample for leishmaniasis proved positive using PCR but negative by direct examination and culture. Treatment was initiated with meglumine antimoniate. A further cutaneous biopsy revealed diffuse lymphocytic proliferation and led to a diagnosis of cutaneous CD8+ aggressive, epidermotropic, cytotoxic T-cell lymphoma. A PET scan showed multiple sites of hypermetabolism affecting the face and lymph nodes. Meglumine antimoniate was stopped and the patient experienced complete remission after chemotherapy. CONCLUSION: Ulcerated nodules with acute progression on acral sites are characteristic of cutaneous CD8+ aggressive, epidermotropic, cytotoxic T-cell lymphoma. In our case, the positive result of PCR screening for Leishmania that was ultimately considered a false positive was a confounding factor in the diagnostic process. Regarding therapy, aggressive treatment strategies such as multiagent chemotherapy and hematopoietic stem-cell transplantation are needed due to the rapid progression of the lymphoma.

Non-invasive positive-pressure ventilation with positive end-expiratory pressure counteracts inward air leaks during preoxygenation: a randomised crossover controlled study in healthy volunteers.

BACKGROUND: During preoxygenation, the lack of tight fit between the mask and the patient's face results in inward air leak preventing effective preoxygenation. We hypothesized that non-invasive positive-pressure ventilation and positive end-expiratory pressure (PEEP) could counteract inward air leak. METHODS: Healthy volunteers were randomly assigned to preoxygenated through spontaneous breathing without leak (SB), spontaneous breathing with a calibrated air leak (T-shaped piece between the mouth and the breathing system; SB-leak), or non-invasive positive inspiratory pressure ventilation (inspiratory support +6 cm H2O; PEEP +5 cm H2O) with calibrated leak (PPV-leak). The volunteers breathed through a mouthpiece connected to an anaesthesia ventilator. The expired oxygen fraction (FeO2) and air-leak flow (ml s-1) were measured. The primary end point was the proportion of volunteers with FeO2 >90% at 3 min. The secondary end points were FeO2 at 3 min, time to reach FeO2 of 90%, and the inspiratory air-leak flow. RESULTS: Twenty healthy volunteers were included. The proportion of volunteers with FeO2 >90% at 3 min was 0% in the SB-leak group, 95% in the SB group, and 100% in the PPV-leak group (P<0.001). At 3 min, the mean [standard deviation (sd)] FeO2 was 89 (1)%, 76 (1)%, and 90 (0)% in the SB, SB-leak, and PPV-leak groups, respectively (P<0.001). The mean (sd) inward air leak was 59 (12) ml s-1 in the SB-leak group, but 0 (0) ml s-1 in the PPV-leak group (P<0.001). CONCLUSIONS: Preoxygenation through non-invasive positive-pressure ventilation and PEEP provided effective preoxygenation despite an inward air leak. CLINICAL TRIAL REGISTRATION: NCT03087825.

[Pyomyositis in children: Two cases of Staphylococcus aureus infection]. / Pyomyosite de l'enfant : deux cas d'infection à Staphylococcus aureus.

Pyomyositis is a term used to denote primary pyogenic infection of a skeletal muscle. It is a rare disease that was first described in immunodeficient patients living in tropical climates. Lately, however, cases involving healthy children have been described in temperate climates. The origin of primary pyomyositis remains unclear, although it is thought to be caused by seeding from transient bacteremia. Onset of disease is insidious, therefore necessitating rapid diagnosis to successfully select appropriate antibiotic therapy and avoid complications. The signs and symptoms of pyomyositis are nonspecific, which highlights the importance of maintaining a broad differential diagnosis including disease entities such as osteomyelitis and septic arthritis. Magnetic resonance imaging is considered the best imaging modality for distinguishing pyomyositis from other potential etiologies. Here, we present two cases of primary pyomyositis of the lower limb muscles in an 8-year-old boy and in an 11-year-old girl.

Influence of enrichment and isolation media on the detection of Campylobacter spp. in naturally contaminated chicken samples.

Investigating Campylobacter epidemiology requires adequate technique and media to ensure optimal culturing and accurate detection and isolation of Campylobacter strains. In the present study, we investigated the performances of three enrichment durations in Bolton broth (0, 24 and 48h) and compared four isolation media (mCCDA, Karmali, Butzler no. 2 and CampyFood agar (CFA)) for the detection of Campylobacter positive samples and the identification of Campylobacter species, from naturally contaminated broiler chicken samples (caeca, neck skin from carcasses, and skin from thighs). We compared our local results to those we obtained with samples from a European survey (caeca and neck skin) and a national survey (neck skin, thigh skin, and breast). Direct plating favored the detection of positive samples highly contaminated by Campylobacter (caeca and neck skin from carcasses) whatever the media. A longer enrichment reduced the rates of Campylobacter recovery except when using Butzler no. 2, more particularly for neck skin which background microflora was less important than in caeca. As a matter of fact, enrichment allowed a higher detection rate of positive samples with low Campylobacter contamination levels (breast, thigh skin), this detection being enhanced when using Butzler no. 2. When comparing the 3 other selective media, CFA was the 2nd most efficient media prior to mCCDA and Karmali. Interestingly, enrichment promoted the growth of Campylobacter coli but this promotion was least with Butzler no. 2 agar. Our study has confirmed the need to adapt the method to the types of samples for improving the detection of Campylobacter and that the method may affect the prevalence of the species.

Prognostic and predictive values of oncogenic BRAF, NRAS, c-KIT and MITF in cutaneous and mucous melanoma.

BACKGROUND: Mutations of BRAF, NRAS and c-KIT oncogenes are preferentially described in certain histological subtypes of melanoma and linked to specific histopathological features. BRAF-, MEK- and KIT-inhibitors led to improvement in overall survival of patients harbouring mutated metastatic melanoma. OBJECTIVES: To assess the prevalence and types of BRAF, NRAS, c-KIT and MITF mutations in cutaneous and mucous melanoma and to correlate mutation status with clinicopathological features and outcome. METHODS: Clinicopathological features and mutation status of 108 samples and of 98 consecutive patients were, respectively, assessed in one retrospective and one prospective study. Clinicopathological features were correlated with mutation status and the predictive value of these mutations was studied. RESULTS: This work identified significant correlations between BRAF mutations and melanoma occurring on non-chronic sun-damaged skin and superficial spreading melanoma (P < 0.05) on one hand, and between NRAS mutations and nodular melanoma (P < 0.05) on the other hand. Younger age (P < 0.05), microscopic (P < 0.05) and macroscopic (P < 0.05) lymphatic involvement at diagnosis of primary melanoma were significantly linked to BRAF mutations. A mutated status was a positive predictive factor of a response to BRAF inhibitors (OR = 3.44). Mutated melanoma showed a significantly (P = 0.038) higher objective response rate to cytotoxic chemotherapy (26.3%) than wild-type tumours (6.7%). CONCLUSION: Clinical and pathological characteristics of the primary melanoma differed between wild-type and BRAF- or NRAS-mutated tumours. Patients with BRAF-mutated tumours were younger at diagnosis of primary melanoma. Patients carrying mutations showed better responses better to specific kinase inhibitors and interestingly also to systemic cytotoxic chemotherapy.

Cutaneous melanoma in patients treated with tumour necrosis factor inhibitors: a retrospective series of 15 patients.

BACKGROUND: Several case reports suggested that tumour necrosis factor-α (TNF) inhibitors might increase the incidence and/or alter the natural course of melanoma towards a more aggressive behaviour. OBJECTIVE: Our objective was to point if history of melanoma in patients exposed to TNF inhibitors could present with a particular pattern at diagnosis or during follow-up. METHODS: We performed a retrospective multicentre study settled in the West part of France to collect and analyse all cases of patients with melanoma who received anti-TNF therapy. RESULTS: Fifteen cases were included. First, 10 patients (mean age: 55.6 years; sex ratio: 1) had a melanoma diagnosed after TNF inhibitors initiation. The mean duration between initiation of treatment and melanoma was 48.7 months. Two patients died of metastatic disease. Second, four patients had a past history of melanoma before anti-TNF therapy (mean duration of treatment: 10.8 months). None experienced a progression of melanoma disease. Last, one woman had a past history of melanoma before and then developed a second melanoma when exposed to biotherapy. CONCLUSION: Our case series does not reveal a distinct profile of melanoma in the patients exposed to TNF inhibitors. Additional prospective trials including larger number of patient are needed to demonstrate the possible link between biological therapy with TNF inhibitors and development of melanoma.

Probiotics: a new way to fight bacterial pulmonary infections?

Antibiotics, of which Fleming has identified the first representative, penicillin, in 1928, allowed dramatical improvement of the treatment of patients presenting with infectious diseases. However, once an antibiotic is used, resistance may develop more or less rapidly in some bacteria. It is thus necessary to develop therapeutic alternatives, such as the use of probiotics, defined by the World Health Organization (WHO) as "micro-organisms which, administered live and in adequate amounts, confer a benefit to the health of the host". The scope of these micro-organisms is broad, concerning many areas including that of infectious diseases, especially respiratory infections. We describe the rational use of probiotics in respiratory tract infections and detail the results of various clinical studies describing the use of probiotics in the management of respiratory infections such as nosocomial or community acquired pneumonia, or on specific grounds such as cystic fibrosis. The results are sometimes contradictory, but the therapeutic potential of probiotics seems promising. Implementing research to understand their mechanisms of action is critical to conduct therapeutic tests based on a specific rational for the strains to be used, the dose, as well as the chosen mode and rhythm of administration.

Vemurafenib-induced eccrine squamous syringometaplasia.

We report herein a patient treated with vemurafenib for a stage IV melanoma who developed an eruption related to eccrine squamous syringometaplasia (ESS). This entity is a well-described side effect of cytostatic therapies used for malignant neoplasia and is clinically characterized by a symmetric cutaneous eruption composed of papules and vesicles preferentially located on fold and intertriginous areas. It is histologically defined by a squamous metaplasia of eccrine ductal epithelium. ESS represents another skin eruption to be added to the list of cutaneous adverse events associated with vemurafenib, a selective BRAF inhibitor used to treat patients with metastatic melanoma harboring the V600E mutation. The discussion focuses on the pathogenesis of ESS secondary to vemurafenib and on alternative diagnoses.

Outbreak of extended spectrum beta-lactamase-producing Klebsiella pneumoniae in an intensive care unit (Brest).

INTRODUCTION: We had for aim to describe control and investigation of an outbreak caused by a strain of Extended spectrum beta-lactamase producing Klebsiella pneumoniae in intensive care units of the Brest teaching hospital. PATIENTS AND METHOD: The case definition was a patient infected by or carrying the epidemic strain. Control measures and investigations are presented. A case-control study was conducted in the surgical intensive care unit. Each case was matched with two controls based on admission times in the unit. The study focused on diagnostic and therapeutic procedures, and potential contacts with healthcare workers, in this context of cross transmission. RESULTS: Between February and May 2011, nine cases were reported in the surgical ICU and two in the medical ICU. Eighteen controls were matched with the nine surgical ICU cases. Several factors were found to be statistically associated with infection or colonization by the epidemic strain: the surgical block in which patients had been operated and the ward of first hospitalization; the number of trans-esophageal and trans-thoracic echocardiographies, of central venous catheter insertions, and of surgical operations; intubation. The total number of invasive procedures was also found to be statistically higher among cases. CONCLUSION: This study identified factors associated with colonization or infection by the epidemic strain. These factors might have been involved in the transmission tree, and be vulnerable elements for the prevention of nosocomial infections and colonisations, and their epidemic spread.

[Eruptive naevi in epidermolysis bullosa hereditaria patients]. / Nævus éruptifs sur épidermolyse bulleuse héréditaire.

BACKGROUND: EB naevus (EBN) are little-known, atypical, eruptive, pigmented melanocytic lesions that may occur in former sites of bullae occurring in epidermolysis bullosa hereditaria (EBH). We sought to describe the characteristics of such lesions and assess their course. PATIENTS AND METHODS: This was a retrospective, two-centre study in which data was collated from the medical files of patients with EBN. We analyzed the patients' demographical data as well as the clinical, dermatoscopic, pathological features of EBN and their progression. RESULTS: Eight patients were studied: they were principally Caucasian (5/8), with a sex ratio of 1. All variants of EBH were represented and most were recessive (63%). We analysed 22 EBN, all atypical and emerging before the age of 10 years (73%), ubiquitously distributed and measuring greater than 5 cm(2) (25%). Of the 13 EBN subjected to dermatoscopy, 12 exhibited a benign reticular pattern. Four were biopsied, and analysis revealed three common naevi and one lentigo. After a median follow-up of 8 years, the EBN seen were either stable (68%), had regressed (23%) or had disappeared (one case). No cases of melanoma were diagnosed. DISCUSSION: EBN are acquired and atypical pigmented naevi. Sixty-four cases of EBN have been reported in the literature up to date. The dermatoscopic features may be evocative of melanoma (17/23 EBN), but to our knowledge no cases of melanoma at a naevus site have been reported. Recessive transmission of EBH appears to be a risk factor (63% of cases), a finding supported by certain pathophysiological hypotheses. CONCLUSION: EBN present atypical clinical and dermatoscopic features. However, while prophylactic total excision did not appear warranted in the absence of any reported cases of melanoma, regular clinical follow-up is recommended.

Are physical performance and injury risk in a professional soccer team in match-play affected over a prolonged period of fixture congestion?

In this study, the effects of a prolonged period of fixture congestion (8 successive official matches in 26 days) on physical performance and injury risk and severity in a professional soccer team were investigated. Computerised motion-analysis was used to analyse the overall distance covered and that run at light- (0.0-11.0 km·h - 1); low- (11.1-14.0 km·h - 1); moderate- (14.1-19.7 km·h - 1) and high-intensities (≥19.8 km·h - 1) for the team as a whole. Distances were measured in metres per minute. Information on match injuries was recorded prospectively. The overall distance covered varied across successive matches (p<0.001) as more distance was run in games 4 and 7 compared to 2 and 3, respectively (126.6 ± 12.3 m·min - 1 and 125.0 ± 13.2 m·min - 1 vs. 116.0 ± 8.0 m·min - 1 and 115.5 ± 11.0 m·min - 1). Distance run in light-intensity exercise also varied (p<0.001) as more distance was covered in game 4 vs. 1, 2, 3, 5 and 6 (75.5 ± 3.8 m·min - 1 vs. 70.6 ± 2.4 m·min - 1, 71.8 ± 3.4 m·min - 1, 69.3 ± 2.6 m·min - 1, 71.5 ± 3.1 m·min - 1, and 70.3 ± 2.8 m·min - 1) and in game 8 vs. game 3 (73.1 ± 3.8 vs. 69.3 ± 2.6 m·min - 1), respectively. When comparing match halves, there were no differences across games in overall or high-intensity distance covered and performance in these measures was similar for matches played before, during and after this period. Globally, no difference over the 8 games combined was observed between the reference team and opponents in any of the performance measures whereas the overall distance covered and that in low- (both p<0.001) and high-intensity running (p=0.040) differed in individual games. The incidence of match injury during the congested fixture period was similar to rates reported outside this period but the mean lay-off duration of injuries was substantially shorter during the former (p<0.05). In summary, while the overall distance run and that covered at lower intensities varied across games, high-intensity running performance and injury risk were generally unaffected during a prolonged period of fixture congestion. These results might be linked to squad rotation and post-match recovery strategies in place at the present club.

The effect of a cold environment on physical activity profiles in elite soccer match-play.

In this study, the effect of cold temperature on the physical activity profiles of professional soccer players in official match-play was investigated. Computerised motion-analysis was used to analyse performance in 9 players (4 defenders and 5 midfielders) in 4 temperature ranges: ≤ 5°C (29 matches), 6-10°C (62 matches), 11-20°C (48 matches) and ≥ 21°C (27 matches). Performance was analysed per minute for total distance covered and distance run in 3 categories: 0.0-14.3 km/h (walking/low/moderate); 14.4-19.7 km/h (running); ≥ 19.8 km/h (high-intensity). Results showed that while total distance run per minute was unaffected in colder conditions, midfielders ran significantly shorter distances in warmer temperatures: ≥21°C = 118.7 ± 6.9 m vs. ≤ 5°C=124.2 ± 7.1 m, p < 0.01; 6-10°C = 123.6 ± 6.8 m, p < 0.01; and, 11-20°C = 123.4 ± 5.4, p < 0.05). The total distance covered at 3 intensities and across halves was unaffected by temperature. Similarly, high-intensity efforts across match halves and in the first and final 5-min periods in each half of normal time were unaffected by temperature. In contrast, high-intensity efforts in midfielders across 15-min intervals were affected by temperature with greater distances covered per minute in the 30-45 min period in matches played in temperatures ≤ 5°C vs. the corresponding period in those played in temperatures ≥ 21°C (9.1 ± 3.8 m vs. 6.2 ± 3.0 m, p < 0.05). The present findings generally suggest that physical performance in professional soccer does not decrease in cold temperatures.

[Strontium ranelate-induced DRESS syndrome]. / Syndrome d'hypersensibilité médicamenteuse (DRESS) au ranélate de strontium.

INTRODUCTION: Drug reaction with eosinophilia and systemic symptoms (DRESS) syndrome is a severe form of adverse drug reaction. Strontium ranelate has recently been authorised for postmenopausal osteoporosis. We report a case of strontium ranelate-induced DRESS complicated by linear Ig A dermatosis due to vancomycin. CASE REPORT: A 77-year-old woman with osteoporosis had been treated by strontium ranelate for 4 weeks when she developed a febrile generalized skin rash. Blood tests showed eosinophilia (12.74 × 10(9)/L) and liver damage. A diagnosis of DRESS was made, leading to discontinuation of strontium ranelate and prescription of systemic corticosteroids. Two days later, methicillin-resistant Staphylococcus aureus bacteraemia occurred and treatment with vancomycin was started. The liver dysfunction resolved. After two weeks of antibiotherapy, bullous lesions were noted on the thighs. Skin biopsy results suggested a diagnosis of linear IgA bullous dermatosis. Vancomycin was stopped. Two weeks later, the eruption resolved. The eosinophil count gradually returned to normal after four months of corticosteroid therapy. DISCUSSION: More than 15 cases of DRESS syndrome have been reported in Europe, including 2 deaths related to ranelate strontium, prompting European health authorities to publish a warning concerning the risk of strontium ranelate-induced DRESS. A particular feature of our patient was complication with linear IgA bullous dermatosis caused by vancomycin. In conclusion, it is essential to be aware of the risk of severe cutaneous reaction to strontium ranelate, a new drug used to treat osteoporosis.

[Staphylococcal tracheitis and toxic shock syndrome in a child]. / Laryngotrachéite à staphylocoque doré compliquée d'un syndrome de choc toxique.

Bacterial tracheitis should be raised as a possible cause in cases of severe febrile dyspnea. It can be life-threatening due to airway obstruction or, less frequently, due to toxic shock syndrome. We report the observation of toxic shock syndrome-complicated bacterial tracheitis in a 14-year-old boy. We describe the signs and symptoms of these conditions as well as the principles of treatment.