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PubMed2007

[Multifocal infection due to Mycobacterium intracellulare: first case of interferon gamma receptor partial dominant deficiency in tropical French territory].

Nontuberculous mycobacterial infections are rare in immunocompetent children, and usually present as adenitis. We report a case of a 6-year-old girl with a multifocal chronic osteomyelitis and pulmonary localisation due to Mycobacterium intracellulare associated with an autosomal dominant mutation of interferon gamma receptor 1 gene (INFGR1) leading to a syndrome of mendelian predisposition to mycobacteria infections by partial deficiency of intracellular signalisation of gamma interferon. This child has been cured with anti-mycobacteria drugs and gamma interferon. This report focus on the importance of looking for a susceptibility of the host to infectious diseases, which can lead to a specific treatment. As far as we know, this is the first case described in a tropical area.

Child↗

PubMed2005

Prognosis and survival after radical resection of primary adenosquamous lung carcinoma.

OBJECTIVE: In order to evaluate the follow-up study of surgical treatment for primary adenosquamous lung carcinoma (ASC) we specified prognostic criteria, also in comparison with primary adenocarcinoma (AC). METHODS: The study group consisted of 96 patients discharged between 1990 and 1999 after radical surgical treatment for ASC-80 (83%) men and 16 (17%) women aged 34-73, mean 56 years. Consequently, we evaluated 252 patients operated during the same time period for primary AC. RESULTS: Apart from grading, we did not find any significant differences between both ASC and AC groups of patients. Among the 96 patients operated radically for ASC median overall survival (OS) was 20 months. The cumulative postoperative survival rates at 5 and 10 years were 25.4 and 19.2%, respectively. By comparison, median OS for 252 patients with AC, discharged after surgical treatment in the same period, was 28.5 months and the cumulative postoperative survival rates at 5 and 10 years were 42.5 and 39.1%, respectively (P=0.006). At pathologic stages IA, the cumulative survival rate at 5 years was 63.3% for patients with ASC as compared with 72.1% for patients with AC (P=0.330). However, out of IB stage patients treated surgically for ASC 31.8% survived 5 years in comparison with 56.3% operated for AC (P=0.017). Study of survival rates did not differ significantly between ASC and AC patients at stage IIA (P=0.824) and stage IIB (P=0.217), respectively. Univariate analysis revealed that six factors of tumor size, T status, N status, as well visceral pleura involvement, tumor localization (central vs. peripheral) and tumor structure were significantly associated with the survival rate according to these variables. Multivariate analysis using Cox's proportional hazards model indicated that T factor, nodal involvement and one of the tumor components predominating were significant factors associated with the postoperative survival of patients with ASC. CONCLUSIONS: Our findings indicate that in patients after radical operation for ASC, predominance for one of the histopathological components (adenous or squamous) within primary tumor is attended by worst prognosis. Our study confirmed also that the prognosis of ASC of the lung was poorer than that of primary AC. Lack of generally accepted diagnostic criteria and unclear prognosis, even in the pathologic stage I suggest that there is a need for prospective studies in this respect.

Adenocarcinoma↗

PubMed2004

Nuclear factor kappaB essential modulator-deficient child with immunodeficiency yet without anhidrotic ectodermal dysplasia.

BACKGROUND: Amorphic mutations in the X-linked nuclear factor kappaB essential modulator ( NEMO ) gene cause Incontinentia pigmenti, which is lethal in hemizygous male patients. Hypomorphic NEMO mutations in male patients lead to anhidrotic ectodermal dysplasia (EDA) with immunodeficiency. OBJECTIVE: To report the clinical features of a child bearing a NEMO mutation who displayed an immunodeficiency without EDA. METHODS: Documentation of clinical care, chart review, standard immunologic and microbiological laboratory techniques, mutation analysis of the NEMO gene. RESULTS: Since the age of 15 months, the patient had Mycobacterium avium disease, beginning with multiple adenitis, later followed by disseminated osteomyelitis and dermatitis. In addition, Haemophilus influenzae and Streptococcus pneumoniae infections led to bronchiectasis. An immunologic work-up revealed a low production of IFN-gamma by PBMCs associated with a hyper-IgM phenotype. Despite treatment using repeated cycles of a 4-drug antimycobacterial regimen, continuous subcutaneous IFN-gamma, repeated antibiotic treatment, and intravenous immunoglobulin substitution, the boy remained chronically ill. At the age of 12 years, the disease was complicated by severe autoimmune hemolytic anemia and eventually fatal herpes simplex virus 1 encephalitis despite high-dose acyclovir therapy. Although he did not present any sign of EDA, a novel type of disease-causing hypomorphic NEMO mutation (110-111insC in exon 2) was identified. CONCLUSION: This case demonstrates that patients hemizygous for NEMO mutations can present with an immunodeficiency without EDA. An investigation of NEMO should thus be undertaken in selected children with immunodeficiency despite the lack of EDA.

Carrier Proteins↗

PubMed2006

Macrophage activation syndrome: an autopsy case of sudden death.

In a context of viral gastroenteritis, we report an unusual case of sudden death in an 8-year-old child. The only macroscopic abnormality observed in the autopsy was a diffuse mesenteric adenitis. Organ samples were taken for histopathological examination and a diffuse lymphocytic infiltration was observed. A sinusoidal histiocytic hyperplasia was found in the lymph nodes. Microscopic examination of the lungs and the lymph nodes revealed haemophagocytic lesions (lymphocytes within the cytoplasm of histiocytic macrophages). Immunohistochemical studies demonstrated that the histiocytes were CD68+ PS100- CD1A-. Following this microscopic examination macrophage activation or haemophagocytic syndrome was diagnosed. The syndrome is a distinct clinical entity characterised by fever, pancytopaenia, splenomegaly, and haemophagocytosis in the bone marrow, liver and lymph nodes. It is a clinical entity that is very difficult to diagnose due to the lack of specific clinical signs. It is generally a complication of an infectious process, an aggravation of an auto-immune disease or a complication of a neoplastic process. The physiopathology involves a disregulation of T lymphocytes and particularly T helper lymphocytes. To make this diagnosis the anatomopathological examination must be performed by an experienced practitioner. The presence of a lymphocyte infiltrate of macrophagic histiocytes in myeloid organs and especially positive CD68+ immune markers are the anatomopathological proofs of diagnosis. The autopsy examination must be carefully performed and include systematic sample harvesting for anatomopathological examination. The results of all these examinations taken together allow the diagnosis of haemophagocytic syndrome to be finally made.

Autopsy↗

PubMed2005

Auramine orange stain with fluorescence microscopy is a rapid and sensitive technique for the detection of cervical lymphadenitis due to mycobacterial infection using fine needle aspiration cytology: a case series.

OBJECTIVE: We sought to evaluate the effectiveness of the auramine orange (AO) stain in diagnosing mycobacterial cervical adenitis (MCA) from fine needle aspiration (FNA) cytology. METHODS: A retrospective review of 19 patients evaluated at 2 urban hospitals from 2000 to 2003 for suspected MCA. FNA specimens were inoculated to culture media and had direct smears stained by the auramine acid fast method. RESULTS: Mycobacteria were identified in 16 (84.2%) of 19 AO-stained FNA specimens, with results available within 4 hours. Corresponding cultures were positive for mycobacteria in 12 specimens, 9 tuberculous and 3 nontuberculous, and grew Mycobacterium tuberculosis from the 3 AO-negative specimens. Three of the 4 patients with negative cultures had previously taken anti-mycobacterial medications. CONCLUSION: The AO stain with fluorescence microscopy is a sensitive and rapid method for detecting tuberculous and nontuberculous mycobacteria. It is a valuable tool for the otolaryngologists and pathologists in the diagnosis of MCA.

Adult↗

PubMed2006

The PFAPA syndrome in oral medicine: differential diagnosis and treatment.

Periodic fever syndromes in children can present with orofacial manifestations which contribute to the formulation of a differential diagnosis. Because of the early onset of oral lesions in these syndromes, the oral medicine practitioner may be required to assist in their diagnosis and management. The periodic fever, aphthous stomatitis, pharyngitis and adenitis (PFAPA) syndrome has been classified as a type of periodic fever syndrome ocurring in early childhood. This article presents a review of the published literature on this entity and summarizes current therapeutic options.

Analgesics↗

PubMed2005

Cellular immunophenotyping of exfoliative dermatitis in canine leishmaniosis (Leishmania infantum).

Lymphocyte subsets, major histocompatibility complex (MHC)-II expressing cells and number of amastigotes in the epidermis and dermis were investigated immunohistochemically in 48 dogs with patent leishmaniosis, with or without exfoliative dermatitis (ED) to study the immunopathogenesis of this common cutaneous form of the disease. Skin biopsies were obtained and compared for ED sites (group A, n = 26), normal-appearing skin from the same animals (group B, n = 24), and leishmanial dogs not exhibiting ED (group C, n = 22), and normal controls (group D, n = 22). The CD3+, CD45RA+, CD4+, CD8+ (CD8a+), CD21+, and MHC-II+ cells and leishmania amastigotes were identified immunohistochemically and counted with the aid of an image analysis system. Pyogranulomatous to granulomatous dermatitis, expressed in various histopathological patterns, was noticed in all groups A and B and in half of group C dogs. In the epidermis, the low number of T-cells and their subsets did not differ significantly between groups A and B, but CD8+ outnumbered CD4+ lymphocytes in both groups. MHC-II+ expression on epidermal keratinocytes was intense in the skin with and without lesions from dogs with ED but not in group C dogs. CD3+, CD8+ and MHC-II+ cells were fewer in group C compared to group A and B dogs. In the dermis, CD3+ cells in group A animals were mainly represented by the CD8+. CD45RA+ and CD21+ cells were also seen in high numbers. MHC-II expression, potentially in lymphocytes, fibroblasts, dendritic cells, and macrophages was intense. The numbers of all cellular subpopulations in the dermis were significantly different between the groups, being highest in group A and lowest in group D. In sebaceous adenitis sites, CD4+ outnumbered CD8+ cells in contrast to the neighbouring dermis and the epidermis. The number of CD21+ and CD45RA+ cells was much lower in the inflamed sebaceous glands compared to the dermis. Finally, the number of amastigotes in the normal-appearing skin was significantly higher in the ED dogs (group B) than in those not exhibiting this cutaneous form of the disease (group C).

Animals↗

PubMed1999

Unusual manifestations of invasive pneumococcal infection.

Unusual pneumococcal infections occurred frequently in the preantibiotic age but rapidly declined with the advent of the antibiotic era. Unfortunately, the morbidity and mortality associated with invasive pneumococcal disease remain high despite antibiotic therapy and monumental advances in medical technology. The incidence of invasive pneumococcal disease has increased recently because of the onset of the human immunodeficiency virus (HIV) epidemic and the emergence of antibiotic-resistant pneumococcus. Robert Austrian described the clinical triad of pneumococcal pneumonia, meningitis, and endocarditis, a syndrome that now bears his name. Although seen infrequently today, unusual manifestations of pneumococcal infection such as those Austrian reported still occur. A review of these cases is warranted because, as drug-resistant organisms continue to emerge worldwide, more unusual pneumococcal infections will be seen. Streptococcus pneumoniae is responsible for a remarkable array of disease processes; our literature review uncovered 95 different types of unusual pneumococcal infections representing 2,064 cases. Examples of these infections included pancreatic and liver abscesses, aortitis, gingival lesions, phlegmonous gastritis, inguinal adenitis, testicular and tubo-ovarian abscesses, and necrotizing fasciitis. We also reviewed predisposing underlying illnesses and conditions. Alcoholism, HIV infection, splenectomy, connective tissue disease, steroid use, diabetes mellitus, and intravenous drug use remain common risk factors for invasive pneumococcal infections. Currently, multidrug-resistant S. pneumoniae remains susceptible to vancomycin and several new third-generation fluoroquinolones. As what some fear will be a possible postantibiotic era approaches, clinicians must be able to recognize and manage unusual pneumococcal infections.

Drug Resistance, Microbial↗

PubMed2000

Osteopontin expression correlates with clinical outcome in patients with mycobacterial infection.

Osteopontin (OPN) is a protein that is expressed in chronic inflammatory diseases including tuberculosis, and its deficiency predisposes to more severe mycobacterial infections in mice. However, no reports have identified altered OPN expression in, or correlated these alterations to, infections in humans. The data presented herein identify alterations in the tissue expression of OPN protein and describe an inverse correlation between these levels and disease progression after inoculation of Mycobacterium bovis bacillus Calmette-Guérin vaccine in humans. Patients with regional adenitis and good clinical outcomes had abundant OPN in infected lymph nodes. This pattern of OPN accumulation was also observed in patients infected by M. avium-intracellulare. In contrast, patients with disseminated infection and histologically ill-defined granulomas had no significant osteopontin accumulation in infected lymph nodes; these patients had either deficiencies in the interferon-gamma receptor 1 or idiopathic immune defects. The level of OPN protein expression was inversely correlated with disseminated infection and, of particular interest, with death of the patient. We conclude that osteopontin expression correlates with an effective immune and inflammatory response when humans are challenged by a mycobacterial infection and that osteopontin contributes to human resistance against mycobacteria.

Antigens, CD↗

PubMed2003

Video-assisted mediastinoscopy: experience from 240 consecutive cases.

BACKGROUND: We report our experience with video-assisted mediastinoscopy. METHODS: We retrospectively reviewed clinical records of all patients who underwent video-assisted mediastinoscopy in a 26-month period. Video-assisted mediastinoscopy was performed in the presence of enlarged lymph nodes (short axis > 1 cm) found at computed tomography scan. Data about operative time, node stations sampled, number of biopsies, and operative complications were collected. Results of the pathologic examination were recorded, as well as (when different) the definitive diagnosis. RESULTS: Video-assisted mediastinoscopy was performed in 240 consecutive patients. In 2 patients, the technique was employed for resection of a mesothelial cyst. In the other cases, it was used for diagnosis of enlarged nodes or staging of lung cancer. Mean number of biopsies was 6.0; mean number of sampled nodal stations was 2.3. Mean operative time was 36.6 minutes. Two operative complications occurred: a pneumothorax not requiring drainage and an injury to the innominate artery requiring manubrial split and suture. In 192 patients, the definitive diagnosis was lung cancer (18 small-cell lung cancers). In the remaining 46 patients, video-assisted mediastinoscopy allowed establishment of the diagnosis (sarcoidosis, n = 22; reactive hyperplastic lympho-adenitis, n = 13; tuberculosis, n = 4; involvement by malignancies other than lung cancer, n = 7). Among the 174 patients with non-small cell lung cancer, mediastinal nodal involvement was recognized in 107 cases (N3, n = 28; N2, n = 79). Sixty-seven patients were staged N less than 2; 47 underwent thoracotomy. Postthoracotomy staging agreed with video-assisted mediastinoscopy staging in 44 cases (93.6%). CONCLUSIONS: Video-assisted mediastinoscopy proved to be safe and effective in nodal assessment of the mediastinum.

Adult↗

PubMed1996

Expression of interleukin-1 beta, interleukin-6 and tumour necrosis factor alpha by macrophages in canine lymph nodes with mineral-associated lymphadenopathy, granulomatous lymphadenitis or reactive hyperplasia.

The distribution of CD3+ T lymphocytes and the expression of interleukin-1 beta (IL-1 beta), interleukin-6 (IL-6) and tumour necrosis factor alpha (TNF alpha) by macrophages in canine lymph nodes with mineral-associated lymph-adenopathy, granulomatous lymphadenitis or reactive hyperplasia was examined immunohistochemically. In areas of preserved lymph node architecture, CD3+ T lymphocytes were located in the paracortex, with scattered cells in the follicular mantle and germinal centre, and small aggregates in the medullary cords. T cells were prominent within areas of diffuse granulomatous inflammation and formed a peripheral rim around microgranulomas. TNF alpha expression was demonstrated in macrophages from seven of 11 dogs with mineral-associated lymphadenopathy, one of five dogs with granulomatous lymphadenitis and three of five with reactive hyperplasia. Positive macrophage staining for IL-1 beta and IL-6 was seen in two lymph nodes with reactive hyperplasia. Lymph nodes from control (clinically normal) dogs showed no evidence of cytokine expression. Neutrophils expressing IL-1 beta were observed within the medullary sinuses of two lymph nodes with mineral-associated lymphadenopathy and one with reactive hyperplasia, and in the inflammatory infiltrate in one lymph node with granulomatous lymph-adenitis. The sensitivity of immunohistochemical examination as a means of detecting cytokines in abnormal tissue is discussed.

Animals↗

PubMed1979

Management of atypical mycobacterial lymphadenitis in childhood: a review based on 380 cases.

The medical and surgical therapy of 82 cases of atypical mycobacterial adenitis from Dallas and 298 cases from the literature was reviewed. The 92% cure rate in 149 patients with total surgical excision alone was comparable to the 95% cure rate in 156 patients when excision was followed by antituberculous drug therapy. With incision and drainage in 63 patients the cure rate was 16% whether drugs were given or not. Ten patients were initially treated with antituberculous drugs alone and only one was cured. It is concluded that total surgical excision is definitive therapy for this disease and that antituberculous drugs should be used only when surgery cannot be performed or when complete excision is not possible.

Antitubercular Agents↗

PubMed1988

Standardized symptomatic treatment versus penicillin as initial therapy for streptococcal pharyngitis.

A multicenter, double-blind, randomized, placebo-controlled trial was conducted to determine whether the addition of penicillin was superior to patient education and anti-inflammatory drug therapy for relief of the acute discomforts of pharyngitis caused by group A beta-hemolytic streptococcus (GABHS). One hundred seventy-eight patients, aged 4 to 29 years, received appropriate symptomatic therapy, including specific doses of aspirin or acetaminophen, plus penicillin (91 patients) or placebo (87) for the initial 48 hours of illness. All had 24-hour office and 48-hour telephone reevaluations. In 123 patients (57 with clinically severe pharyngitis), throat cultures yielded GABHS. Penicillin provided a margin of 20% improvement over anti-inflammatory therapy for the complaint of sore throat only after 48 hours of treatment (for the 123 patients with GABHS, p = 0.01; for the 57 with both severe pharyngitis and GABHS, p = 0.05). No significant improvement was noted for fever, malaise, odynophagia, exudate, adenitis, or pharyngitis. The failure of penicillin to provide much additional benefit makes its routine early prescription specifically for symptomatic relief questionable.

Acetaminophen↗

PubMed1979

Yersinia enterocolitica infections in children.

Y. enterocolitica has been increasingly associated with a wide range of age-related clinical manifestations in children and adults, including febrile gastroenteritis, pseudoappendicitis, arthritis, sepsis, and focal suppurative disease. Although definite patterns of incidence, prevalence, transmission, and pathophysiology are emerging, much remains to be explained. The alert clinician who notifies his clinical laboratory colleagues that special isolation techniques are required to recover this organism from stool samples, and who submits mesenteric lymph nodes for bacteriologic examination in cases of mesenteric adenitis, will aid attempts to further delineate the significance of this emerging pathogen in the United States. Therapy depends on the form and severity of illness and must be guided by in vitro sensitivity, pending animal and epidemiologic studies.

Animals↗

PubMed1977

Differential tuberculin test for mycobacterial infection in children.

A composite antigen prepared from strains of M. avium, M. intracellulare and M. scrofulaceum has been used in addition to PPD in differential skin tests, principally on children investigated for mycobacterial cervical adenitis. The tests were uniformly successful in 12 cases bacteriologically positive for opportunist mycobacteria and gave unequivocal positive or negative results in 30 cases awaiting diagnosis.

Antigens, Bacterial↗

PubMed1984

Nontuberculous mycobacterial infections: comparison of the fluorescent auramine-O and Ziehl-Neelsen techniques in tissue diagnosis.

Nontuberculous (i.e., atypical) mycobacterial infections are increasing among pediatric and immunosuppressed patients, who commonly present with subcutaneous inflammatory masses or adenitis, which is often surgically excised. Since the most frequently isolated species also grow slowly in culture, early diagnosis may depend on histologic detection of a mycobacterial organism in the biopsy specimen. However, the histologic methods used for this purpose are of uncertain value in the diagnosis of these infections. Biopsy specimens from 22 patients with clinical histories highly consistent with nontuberculous mycobacteriosis in which part of the tissue was cultured were selected for study. Coded tissue blocks and control specimens were stained by the Ziehl-Neelsen (ZN) or auramine-O (A0) fluorescent technique and examined blindly for the presence of characteristic organisms. Results of these studies were compared with the culture results, and predictive values were calculated. This experience showed that the AO technique is technically simpler, allowing faster screening at lower power and showing greater sensitivity and predictive value of a negative result although less specificity than the ZN technique. The lower specificity of AO may be factitious and due to the detection in the tissue of organisms that did not grow in culture. Previous observations that nontuberculous mycobacterial infections may elicit tissue reactions that simulate cat-scratch disease, sarcoidosis, and nonspecific chronic inflammation were also confirmed.

Adult↗

PubMed1975

Appendicitis and mimicking conditions. A prospective study.

A prospective study of 444 consecutive patients diagnosed as having acute appendicitis was carried out in a district general hospital. The appendix was acutely inflamed, gangrenous, or perforated in 346 patients. Diagnostic error, 22% overall, was twice as common in females as in males. Organisms were isolated from the outer appendix wall in 117 patients, isolation increasing with the severity of inflammation. 12% of children under 11 had mesentric adenitis, 10% of all females had gynaecological lesions, and 14% of patients over 50 had acute diverticulitis. In only 6% of patients was no abnormality found at operation, and in every case the disorder was dealt with through the gridiron incision.

Abdomen, Acute↗

PubMed1982

Toxic-shock syndrome not associated with menstruation. A review of 54 cases.

An increasing proportion of reported cases of toxic-shock syndrome (TSS) are not associated with menstruation (13.2% of reported cases with onset in 1981). The 54 cases of TSS not associated with menstruation reported through the U.S.A. national surveillance system between January, 1980, and June, 1981, were either associated with Staphylococcus aureus infections (cutaneous or subcutaneous lesions, infected surgical wounds, bursitis, mastitis, adenitis, lung abscess, or primary bacteraemia) or followed childbirth by vaginal delivery and caesarean section. Patients with TSS not associated with menstruation differed significantly in age and racial distributions from those with menstruation-associated TSS, and 17 of them were male. The clinical features of TSS not associated with menstruation and the characteristics of the S. aureus strains isolated from these patients were similar to those observed in TSS related to menstruation. The median incubation period of the disease in the post-surgical cases was 2 days. TSS can occur in many clinical settings in patients of both sexes and of all ages and racial groups.

Adolescent↗