Anne-Sophie Morin

Tuberculous cerebral vasculitis: Retrospective study of 10 cases

BACKGROUND Tuberculous cerebral vasculitis is a complication of tuberculous meningitis. This study was undertaken to determine the epidemiological characteristics, context, diagnostic means and outcomes under treatment of tuberculous cerebral vasculitides. METHODS All consecutive patients diagnosed with tuberculous cerebral vasculitis were identified from the databases of three Internal Medicine, one Neurology and one Infectious Disease Departments in three suburban Parisian hospitals. RESULTS We describe 10 cases: five men and five women (median age 33.5 [range: 27-55] years). Two were infected with the human immunodeficiency virus. Nine patients had tuberculous meningitis, eight with extraneurological involvement. The following manifestations led to the diagnosis: motor deficit, acute confusional state, headaches, involvement, coma and/or seizures. The cerebral vasculitis revealed tuberculosis in three patients, but tuberculosis was already known when vasculitis was diagnosed for the seven others. The cerebral computed-tomography scan showed cerebral infarctions in five patients, hydrocephalus and tuberculomas in four, while magnetic resonance imaging detected infarctions and leptomeningitis in nine patients, pachymeningitis in one, hydrocephalus and tuberculomas in seven. Therapy combined antituberculous agents with oral corticosteroids for all patients, preceded by a methylprednisolone pulse for five patients. Outcome was favorable for nine patients. CONCLUSION We described the non-negligible frequency of tuberculous cerebral vasculitides, their clinical manifestations and their potential severity, and the diagnostic and monitoring contributions of magnetic resonance imaging and magnetic resonance angiography.

Immune thrombocytopenia in adults: a prospective cohort study of clinical features and predictors of outcome

This prospective observational cohort study aimed to explore the clinical features of incident immune thrombocytopenia in adults and predictors of outcome, while determining if a family history of autoimmune disorder is a risk factor for immune thrombocytopenia. All adults, 18 years of age or older, recently diagnosed with immune thrombocytopenia were consecutively recruited across 21 hospital centers in France. Data were collected at diagnosis and after 12 months. Predictors of chronicity at 12 months were explored using logistic regression models. The association between family history of autoimmune disorder and the risk of developing immune thrombocytopenia was explored using a conditional logistic regression model after matching each case to 10 controls. One hundred and forty-three patients were included: 63% female, mean age 48 years old (Standard Deviation=19), and 84% presented with bleeding symptoms. Median platelet count was 10×109/L. Initial treatment was required in 82% of patients. After 12 months, only 37% of patients not subject to disease-modifying interventions achieved cure. The sole possible predictor of chronicity at 12 months was a higher platelet count at baseline [Odds Ratio 1.03; 95%CI: 1.00, 1.06]. No association was found between outcome and any of the following features: age, sex, presence of either bleeding symptoms or antinuclear antibodies at diagnosis. Likewise, family history of autoimmune disorder was not associated with incident immune thrombocytopenia. Immune thrombocytopenia in adults has been shown to progress to a chronic form in the majority of patients. A lower platelet count could be indicative of a more favorable outcome.

Prolonged paradoxical response to anti-tuberculous treatment after infliximab

A 56-year-old woman with ankylosing spondylitis, treated for 3 months with infliximab, developed miliary tuberculosis with mediastinal lymphadenopathies and brain and splenic lesions. After initial improvement under anti-tuberculous therapy, she suffered an unexpectedly prolonged paradoxical worsening with several episodes of lymphadenopathy, including life-threatening ones, over a period of more than 14 months of follow-up. The outcome was favorable as a result of corticosteroid and surgical treatments. This phenomenon reflects a paradoxical reaction precipitated by infliximab withdrawal.

Vincristine efficacy and safety in treating immune thrombocytopenia: a retrospective study of 35 patients.

Although vincristine (VCR) is sometimes prescribed for newly diagnosed immune thrombocytopenia (ITP), its efficacy in refractory ITP and sustained efficacy has yet to be demonstrated. We describe our clinical experience and recommend vincristines correct place in ITP management. This retrospective study analysed data from 35 patients with newly diagnosed (ND), persistent (P) or chronic (C) ITP treated with VCR. The initial response rate, defined as >30 × 109 platelets/L, reached 86% after a median of 7 [interquartile range (IQR) 6–13] days. In ND and P ITP, even when previous therapies were inefficient, initial response was 87.5%, suggesting that this treatment could be used particularly in rescue. Median survival time, without failure or relapse, was 15 months (Kaplan–Meier curve). Predictive factors (univariate analysis) of an initial and long‐term response were a small number of prior treatments received. However, at 2 yr, only seven patients had sustained response. Eight (23%) patients experienced adverse events: neuropathy for seven and bowel obstruction for one. Vincristine efficacy in ITP was confirmed, and it could be a good strategy for treating resistant ITP, especially in emergencies. In this era of new therapeutics, VCR deserves to remain on the list of ITP treatments because of its initial efficacy, safety and low cost.

Risk factors associated with intracranial hemorrhage in adults with immune thrombocytopenia: A study of 27 cases.

LMW iron dextran over 1 hr. An additional 30-min infusion time was used for each 500 mg beyond the traditional 1 gram dose and premedication use was only recommended with >2 drug allergies. We conducted a retrospective cohort study of adults receiving iron dextran for the first time at a cancer center located on the campus of a regional academic medical center. Patients were divided into shortened infusion (rate 12 mg/min) and extended infusion (rate <12 mg/min) iron dextran groups. Infusion time was based on rounding up to the nearest 500 mg dose. A total of 241 were screened for inclusion, 34 of whom were excluded (20 previously received iron dextran, 6 concurrent chemotherapy, 4 incomplete time information, 3 experienced an AE during test dose, and 1 did not receive iron dextran). Of those who met inclusion, 92 (45%) received the extended infusion and 115 (55%) received the shortened infusion. At baseline, there were no statistically significant differences between groups with the exception of more females (81.5% vs. 67.8%; P 5 0.026) and premedication use (81.5% vs. 46.9%; P< 0.001) in the extended infusion group. The most common indications for parenteral iron were gastrointestinal or uterine bleeding (35%) and cancer (28%). AEs and AEs requiring medication intervention were not significantly different between groups (Table I). The most common AEs were abdominal pain and nausea. Throat itching occurred in two (both in the extended infusion group) and led to infusion discontinuation in one. The univariate analysis of baseline characteristics to identify correlation with AEs only detected a significant association with celiac disease. Celiac disease, as well as those variables that differed significantly at baseline between the two groups (female sex and premedication use), were incorporated into the multiple logistic regression. In that analysis, celiac disease remained the only characteristic significantly associated with increased risk of developing an AE (OR 39.9, 95% CI 3.8–417.4; P 5 0.002). Of the four with celiac disease in the study, three developed an AE. There were also no significant differences between groups with regard to average increase in hemoglobin or any of the iron indices (Table I). Chair time was significantly less, by an average of 1.7 hr, in the shortened infusion group (Table I). The results of this study show that there is no difference in AEs and effectiveness between shortened and extended infusion iron dextran. AEs were relatively uncommon and nonlife threatening. After controlling for gender and premedication use, celiac disease was the only baseline characteristic significantly associated with an increased likelihood of an AE. Although previous studies have not reported a relationship between celiac disease and AEs, it may be related to the immune-mediated nature of the disease. However, this association should be viewed cautiously as there were only four (1.9%) with celiac disease. Notably, premedication use did not significantly reduce the risk of developing an AE. While premedication use did decline significantly after protocol implementation, it is suspected that many continued to receive premedication because of provider comfort. Antihistamines were the most commonly administered premedication, despite the lack of evidence and their ability to cause AEs themselves [4]. While it is difficult to differentiate, it is possible that many of the AEs experienced were secondary to anti-histamine use rather than iron dextran. Those with >2 drug allergies made up a relatively large percentage of the population (12%) but, unlike the previous study, we found no significant association between multiple drug allergies and AEs. This could be explained by premedication use in this group, although this is common practice in many studies and the protective effect of premedication is not convincing [3,5]. Finally, the switch to shortened infusion iron dextran also has the advantage of significantly less chair time. This time savings is beneficial as it allows more to receive treatment and requires less time spent in clinic. Additionally, it improves reimbursement rates, as a clinic is often paid more for the first hour of infusion than subsequent hours. Although several studies have assessed the safety and effectiveness of shortened infusion iron dextran, this study is unique in that it directly compares extended versus shortened infusion in a general population [3,5,6]. Limitations include the retrospective nature and infrequency of severe AEs. Since severe AEs are exceedingly rare, especially with the disappearance of HMW formulations, it is logistically challenging to collect a sample size that would have a high likelihood of capturing such events. Future studies are necessary to further elucidate risk factors associated with developing an AE in order to identify if certain populations may benefit from premedication. In summary, iron dextran is a convenient parenteral treatment option that allows for total iron deficit repletion, which is particularly beneficial for those who live in rural areas or have difficulty getting into clinic. Although historical concerns regarding serious iron dextran AEs have led to standard extended infusion times, the results of this study support the transition to shortened infusion as safe, effective, and associated with significant chair time savings.

Plasmocytose sanguine et médullaire au cours de la dengue : une observation

We report a case of dengue fever with plasma cells in the blood (3980 per cubic millimeter) and bone marrow (30%) in a 55-year-old woman hospitalized for fever, arthralgias and thrombocytopenia (66,000 per cubic millimeter) on returning from the West Indies. Serological testing confirmed the diagnosis. Plasmacytosis is rare in dengue fever and its frequency and correlation with the different forms of the disease remain to be determined.

[Treatment duration of extra-pulmonary tuberculosis: 6 months or more? TB-INFO database analysis].

PURPOSE The recommended duration of pulmonary tuberculosis therapy is 6 months. For extrapulmonary tuberculosis, treatment duration depends on tuberculosis involvement and HIV status. The objective of this study was to describe the main characteristics of a cohort of extrapulmonary tuberculosis patients, to compare patients with a 6-month treatment to those with more than a 6-month treatment, and to analyze the compliance of medical centres with recommended duration of treatment. METHODS A retrospective cohort study of 210 patients with extrapulmonary tuberculosis was carried from January 1999 to December 2006 in two hospitals in the north-east of Paris. These patients were treated with quadruple therapy during two months, followed by dual therapy during 4 months (n=77) or more (n=66). The characteristics of each group were compared by uni- and multivariate analysis. The primary endpoint was the rate of relapse or treatment failure at 24-month follow-up after treatment completion. RESULTS No relapse was observed after 24 months of follow-up after the end of treatment in the two groups. In univariate analysis, patients with lymph node tuberculosis were more often treated for 6 months than at other sites of tuberculosis (respectively 61% versus 40.9%; P=0.02); the decision of treatment duration was related to medical practices (79.2% treated 6 months in one hospital versus 20.7% in the other, P<0.001); patients living in private residence were more often treated during 6 months than patients living in residence (24.2% versus 10.3%, P=0.042). In multivariate analysis, only hospital (P=0.046), sex (P=0.007) and private residence were significantly different in each group. CONCLUSION A period of 6 months seems to be sufficient to treat extrapulmonary tuberculosis (except for neuromeningeal localization).

Ileal leiomyosarcoma and lymph node granuloma. Relevance of a rare association

Leiomyosarcoma is rare in ileal location. We report the case of a 61 years old female patient presenting with ileal leiomyosarcoma occurring at 14 years after a uterine carcinoma treated by radiotherapy. The ileal tumor was treated by surgical resection. This tumor was peculiar by the macroscopic polypoid appearance and by expression of PDGFRA protein together with muscle differentiation proteins: smooth muscle actin, desmin and h-caldesmon. Lymph node necrotizing granuloma diagnosis on the surgical resection specimen lead to the diagnosis of tuberculosis and the patient was treated accordingly. At 3 years after the diagnosis, the patient was well, without recurrence or metastases. In conclusion, we report the case of a patient diagnosed with ileal leiomyosarcoma occurring 14 years after adjuvant radiotherapy for uterine carcinoma. Analysis of the intestinal resection specimen lead to the diagnosis of associated tuberculosis. Moreover, the leiomyosarcoma was peculiar by PDGFRA expression, feature which might be of clinical relevance since the treatment options in radioinduced tumors associated with other conditions are limited.

Nasopalpebral Schwannomas and Human Immunodeficiency Virus Infection.

Objective: To report a case of a schwannoma of nasopalpebral location, occurring in a human immunodeficiency virus (HIV)-positive patient. Clinical Presentation and Intervention: A 55-year-old man presented with a nasopalpebral painless tumefaction, pneumopathy and HIV-related immunodepression after stopping combination antiretroviral therapy. Magnetic resonance imaging showed subcutaneous masses, with contrast enhancement of the left nose pyramid, internal cantus and inferior palpebral area, suspicious of Kaposi sarcoma. The resected specimen showed schwannoma histology, with tumor cells expressing S100 protein and WT1. Conclusion: The features of a rare case of facial schwannoma of nasopalpebral location in an HIV-positive patient are reported. The diagnosis may be difficult before microscopic examination, with imaging features suggesting a Kaposi sarcoma.

Multifocal bone aspergillosis by Aspergillus terreus in an apparently immunocompetent patient.

La Presse Medicale - In Press.Proof corrected by the author Available online since mardi 1 septembre 2015