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AIM: There is no validated symptom scale for Swedish children with gastrointestinal disorders. Our aim was to validate the Swedish version of the Paediatric Quality of Life Inventory (PedsQL) gastrointestinal symptoms scale and symptoms module. METHODS: Families were recruited from two hospitals in Gothenburg, Sweden, from 1 March 2021 to 31 October 2022. The instruments were completed by 115 children with functional, congenital or organic acquired gastrointestinal disorders and 149 of their parents. These were the gastrointestinal symptoms scales, symptoms module and the 4.0 Generic core scale. Data were analysed for feasibility, construct validity and reliability, including internal consistency, re-test reliability and child-parent agreement. RESULTS: Feasibility was good, with a failure to respond of ≤5%. Construct validity showed strong correlation in the PedsQL gastrointestinal symptoms module. The known-group validity agreed with the expectations associated with the disease characteristics (p < 0.05). Cronbach's alpha was 0.96, which indicated excellent internal reliability. The intraclass correlation coefficient for the child self-report and parent-proxy report was 0.74, which indicated good agreement. CONCLUSION: The Swedish PedsQL Gastrointestinal Symptoms Scales, the symptoms module provided acceptable measurement properties and can be used to evaluate symptoms of gastrointestinal disorders and quality of life during clinical work or research projects.
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BACKGROUND: Short-chain fatty acids (SCFAs) in intestinal contents may influence immune function, while less is known about SCFAs in blood plasma. The aims were to investigate the relation between infants' and maternal plasma SCFAs, as well as SCFAs in mother's milk, and relate SCFA concentrations in infant plasma to subsequent sensitisation and atopic disease. METHODS: Infant plasma (N = 148) and corresponding mother's milk and plasma were collected four months postpartum. Nine SCFA (formic, acetic, propionic, isobutyric, butyric, succinic, valeric, isovaleric, and caproic acid) were analysed by UPLC-MS. At 12 months of age, atopic disease was diagnosed by a pediatric allergologist, and sensitisation was measured by skin prick test. All families participated in the Swedish birth cohort NICE (Nutritional impact on Immunological maturation during Childhood in relation to the Environment). FINDINGS: Infants with sensitisation, atopic eczema, or food allergy had significantly lower concentrations of five, three, and two SCFAs, respectively, in plasma at four months. Logistic regressions models showed significant negative associations between formic, succinic, and caproic acid and sensitisation [ORadj (95% CI) per SD: 0.41 (0.19-0.91); 0.19 (0.05-0.75); 0.25 (0.09-0.66)], and between acetic acid and atopic eczema [0.42 (0.18-0.95)], after adjusting for maternal allergy. Infants' and maternal plasma SCFA concentrations correlated strongly, while milk SCFA concentrations were unrelated to both. Butyric and caproic acid concentrations were enriched around 100-fold, and iso-butyric and valeric acid around 3-5-fold in mother's milk, while other SCFAs were less prevalent in milk than in plasma. INTERPRETATION: Butyric and caproic acid might be actively transported into breast milk to meet the needs of the infant, although mechanistic studies are needed to confirm this. The negative associations between certain SCFAs on sensitisation and atopic disease adds to prior evidence regarding their immunoregulatory potential. FUNDING: Swedish Research Council (Nr. 2013-3145, 2019-0137 and 2023-02217 to A-S.S.), Swedish Research Council for Health, Working Life and Welfare FORTE, Nr 2018-00485 to A.W.), The Swedish Asthma and Allergy Association's Research Fund (2020-0020 to A.S.).
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Dermatitis Atópica , Leche Humana , Lactante , Femenino , Humanos , Niño , Leche Humana/química , Caproatos/análisis , Dermatitis Atópica/diagnóstico , Dermatitis Atópica/etiología , Madres , Cromatografía Liquida , Espectrometría de Masas en Tándem , Ácidos Grasos Volátiles/análisis , Ácidos GrasosRESUMEN
OBJECTIVES: The objective of the study is to examine the association between the lack of follow-up for celiac disease (CD) during childhood and dietary adherence, disease remission, and health-related quality of life (HRQoL). METHODS: We invited 243 randomly selected children diagnosed with CD in 2013-2018 in Gothenburg, Sweden, and 162 consented to participate (67%). We retrieved information on clinical follow-up and current wellbeing using medical and laboratory records data, as well as validated questionnaires on symptoms of CD, dietary adherence, and HRQoL. We analyzed tissue-transglutaminase antibodies (tTGA) as a measure of disease remission. We defined lack of follow-up as no CD-related physician/dietician-led visit or measurement of tTGA over the past 24 months of study enrollment. RESULTS: The mean age at study enrolment was 12.7 (range 7.8-18.2) years. Out of 162 children with an average disease duration of 5.3 (range 2.3-8.8) years, 23 (14%) lacked follow-up. tTGA had normalized in 94% [95% confidence interval (CI) = 71%-100%] of children without follow-up versus 91% (95% CI: 85%-95%) of children with continued follow-up. Of children without follow-up, 65% (95% CI: 38%-86%) reported a dietary adherence score indicating very good adherence, versus 72% (95% CI: 63%-80%) of those with continued follow-up. Also, lack of follow-up was not significantly associated with growth, symptom scores, or HRQoL. CONCLUSIONS: In this regional cohort study of mostly older children and adolescents, lack of follow-up for CD was not significantly linked to dietary adherence, disease remission, or HRQoL. How these results hold in larger, unselected samples with longer follow-up, including transition to adult care, warrants further study.
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Enfermedad Celíaca , Niño , Adulto , Adolescente , Humanos , Enfermedad Celíaca/diagnóstico , Enfermedad Celíaca/terapia , Estudios de Cohortes , Suecia/epidemiología , Estudios de Seguimiento , Calidad de Vida , AutoanticuerposRESUMEN
BACKGROUND: Orofacial granulomatosis (OFG) is an inflammatory disorder of the perioral region and oral cavity. Crohn's disease (CD) in conjunction with OFG (CD-OFG), has been suggested to constitute a phenotype of CD with distinct features at diagnosis. AIMS: The aim of this project was to investigate whether the distinct phenotypic features of CD-OFG persist in the years following the initial diagnosis of CD. METHODS: Clinical data were extracted from medical records covering the first 5 years post-diagnosis for a cohort of patients with CD-OFG, and were compared to those of references with CD without OFG. RESULTS: The clinical characteristics of our cohort of patients with CD-OFG (N = 25) were evaluated in comparison to references with CD without OFG (ratio 1:2). Five years post-diagnosis, more patients with CD-OFG had a phenotype with perianal disease (cumulative incidence: 16/25, 64% vs 13/50, 26%, P = 0.002) and intestinal granulomas (cumulative incidence: 22/25, 88% vs 24/50, 48%, P = 0.0009) than patients in the CD reference group. The patients with CD-OFG were also more likely to have undergone perianal surgery (12/25, 48% vs 4/50, 8%, P = 0.0002). At the end of the observation period, more of the patients with CD-OFG were receiving combination therapy, i.e., immunomodulators and tumor necrosis factor antagonists, than those in the CD reference group (9/25, 36% vs 5/50, 10%, P = 0.01). CONCLUSION: The results support the notion that CD in conjunction with OFG represents a specific phenotype of CD that is characterized by frequent perianal disease, pronounced intestinal granuloma formation and a need for extensive therapy.
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Enfermedad de Crohn , Granulomatosis Orofacial , Enfermedades Intestinales , Humanos , Enfermedad de Crohn/complicaciones , Enfermedad de Crohn/diagnóstico , Enfermedad de Crohn/tratamiento farmacológico , Granulomatosis Orofacial/diagnóstico , Granulomatosis Orofacial/tratamiento farmacológico , Granulomatosis Orofacial/epidemiología , Intestinos/patología , Granuloma/epidemiología , Enfermedades Intestinales/patologíaRESUMEN
AIM: To examine the clinical follow up of paediatric coeliac disease and the rate of loss of follow up during childhood, for which data are scarce. METHODS: In a cohort of coeliac children diagnosed in 2013-2018 in Gothenburg, Sweden, we retrospectively explored the follow-up practice of paediatric coeliac disease until June 2021. We used medical records from hospital-based paediatric gastroenterology and general paediatric outpatient clinics, laboratory records, and questionnaires. Loss of follow up was defined no coeliac disease-related follow up or tissue transglutaminase test over the past 2 years of study enrolment. RESULTS: We included 162 children (58% girls) aged 7.8-18.2 years (average 12.7). Most participants (76%) were followed at general paediatric outpatient clinics rather than hospital-based clinics. After 2.3-8.8 (average 5.3) years since diagnosis, 23 patients (14%; 95% confidence interval, 9%-21%) had been lost to follow up. Patients with loss of follow up were more often boys (61% versus 39%, p = 0.08), with a somewhat longer average disease duration of 5.8 versus 5.2 years (p = 0.11). There were no between-group differences in socio-economic characteristics and patient-reported experience measures of coeliac disease care. CONCLUSION: One in seven coeliac patients may experience loss of follow up during childhood.
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Enfermedad Celíaca , Niño , Masculino , Femenino , Humanos , Suecia/epidemiología , Estudios de Seguimiento , Estudios Retrospectivos , Enfermedad Celíaca/epidemiología , Enfermedad Celíaca/diagnóstico , Encuestas y CuestionariosRESUMEN
BACKGROUND: Young adults with childhood-onset inflammatory bowel disease (IBD) have increased risks of low areal bone mineral density and low skeletal muscle mass. Volumetric BMD (vBMD), bone geometry and microstructures, in addition to possible associations with skeletal muscle index (SMI) and physical exercise have been scarcely studied in this patient group. PATIENTS AND METHODS: In total, 49 young adult male patients with childhood-onset IBD and 245 age- and height-matched young adult male controls were scanned with high-resolution peripheral quantitative computed tomography. Bone geometry, vBMD, and bone microstructures were calculated as median values and compared between the patients and controls. Multivariable linear regression analyses were performed to determine the independent associations among IBD diagnosis, SMI (kg/m2), and physical exercise. RESULTS: The group of young adult patients had, in comparison with the controls, significantly smaller median cortical area (126.1 mm2 vs151.1 mm2, Pâ <â .001), lower median total vBMD (296.7 mg/cm3 vs 336.7 mg/cm3, Pâ <â .001), and lower median cortical vBMD (854.4 mg/cm3 vs 878.5 mg/cm3, Pâ <â .001). Furthermore, the patients compared with the controls had lower median trabecular volume fraction (16.8% vs 18.2%, Pâ <â .001) and thinner median trabeculae (0.084 mm vs 0.089 mm, Pâ <â .001). The differences between the patients with IBD and controls persisted in multivariable analyses that included adjustments for SMI and physical exercise. CONCLUSIONS: Young adult men with childhood-onset IBD are at increased risk of having reduced bone quality in both the cortical and trabecular bone structures compared with normative matched controls.
Young adult men with childhood-onset IBD appear to have deficits in both cortical and trabecular bone microstructures, measured with high resolution peripheral computed tomography, compared with age- and height-matched young adult male controls.
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Enfermedades Óseas Metabólicas , Enfermedades Inflamatorias del Intestino , Humanos , Masculino , Adulto Joven , Niño , Densidad Ósea , Absorciometría de Fotón/efectos adversos , Hueso Esponjoso/diagnóstico por imagen , Huesos/diagnóstico por imagen , Enfermedades Óseas Metabólicas/etiología , Enfermedades Inflamatorias del Intestino/complicacionesRESUMEN
BACKGROUND: Patients with inflammatory bowel disease (IBD) have an increased risk of compromised bone mineral density (BMD) and body composition. There are limited data on the physical exercise (PE) habits of patients with childhood-onset IBD and on the associations between PE and BMD and body composition. PATIENTS AND METHODS: In total, 72 young adults with childhood-onset IBD and 1341 normative young adult controls answered questionnaires regarding PE [hours/week (h/w)] in the last 12 months. BMD and body composition were measured with dual x-ray absorptiometry (DXA) and presented as age- and gender-adjusted Z-scores for BMD, skeletal muscle index (SMI, the weight of lean mass in arms and legs/m2), and percentage body fat (Fat %). RESULTS: A total of 41 (57%) patients with IBD engaged in PE during the previous 12 months, as compared to 913 (68%) of the controls (p = .053). Sedentary patients had significantly lower median BMD, SMI, and Fat % Z-scores than the controls with corresponding PE habits (all p < .05). In contrast, highly active (>4 h/week) patients had total body BMD, SMI, and Fat % in the same range as the controls with corresponding PE levels (p = .151, p = .992, and p = .189, respectively), albeit with lower BMDs in the spine (p = .007) and femoral neck (p = .015). Using multiple regression analyses, a diagnosis of childhood-onset IBD was independently associated with inferior BMD and body composition, regardless of the amount of PE. CONCLUSION: Physical exercise is associated with beneficial bone mineral density and body composition in patients with IBD despite the negative effects of the disease.
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Densidad Ósea , Enfermedades Inflamatorias del Intestino , Absorciometría de Fotón , Composición Corporal , Ejercicio Físico , Humanos , Adulto JovenRESUMEN
INTRODUCTION: Collagenous gastritis (CG), a rare disorder of unknown etiology, has been postulated to have immune-mediated mechanisms. We investigated (i) the incidence and prevalence of CG in a pediatric population; (ii) the clinical, endoscopic, and histologic characteristics of childhood-onset CG; and (iii) the evidence for autoimmunity and/or inflammatory activity in these patients. METHODS: Clinical, endoscopic, and histologic data were reviewed longitudinally in a population-based Swedish cohort of 15 patients with childhood-onset CG diagnosed in the period 2008-2019. A set of 11 autoantibodies, 4 blood inflammatory biomarkers, and the human leukocyte antigen DQ2/DQ8 genotype was analyzed cross-sectionally. RESULTS: The incidence rate of childhood-onset CG was 0.25/100,000 person-years, with an incidence rate ratio of girls to boys of 4.2 (95% confidence interval, 1.2-15). The prevalence of CG was 2.1/100,000 in children aged younger than 18 years. The endoscopic and histologic findings remained pathologic in all the examined patients during a median follow-up of 4.4 years. Many patients had heredity for autoimmune disorders (47%) and/or tested positive for autoantibodies (40%) or human leukocyte antigen DQ2/DQ8 (53%). No associated autoimmune comorbidities were observed. The serum levels of calprotectin and amyloid A were increased in 10/15 (67%) and 5/15 (33%) of the patients, respectively, whereas plasma C-reactive protein levels were normal in all, but 1 patient. DISCUSSION: The results indicate that childhood-onset CG is rare and has a chronic disease course. Although signs of autoimmune predisposition are frequent, early development of autoimmune comorbidities seems seldom. Serum calprotectin and amyloid A represent novel candidate biomarkers of inflammatory activity in CG (see Visual Abstract, Supplementary Digital Content 4, http://links.lww.com/CTG/A349).
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Autoanticuerpos/sangre , Colágeno/metabolismo , Mucosa Gástrica/patología , Gastritis/epidemiología , Adolescente , Edad de Inicio , Autoanticuerpos/inmunología , Biomarcadores/sangre , Biopsia , Proteína C-Reactiva/análisis , Niño , Estudios de Cohortes , Estudios Transversales , Femenino , Mucosa Gástrica/inmunología , Gastritis/sangre , Gastritis/inmunología , Gastritis/patología , Antígenos HLA-DQ/sangre , Antígenos HLA-DQ/inmunología , Humanos , Incidencia , Inflamación/sangre , Inflamación/diagnóstico , Inflamación/inmunología , Complejo de Antígeno L1 de Leucocito/sangre , Masculino , Proteína Amiloide A Sérica/análisis , Adulto JovenRESUMEN
Background: Patients with inflammatory bowel disease (IBD) often develop alterations in body composition in terms of their proportions of lean mass and fat mass, as well as reduced bone mineral density (BMD). However, there are limited data on the skeletal muscle index (SMI) and percentage fat (fat %) for young adults with childhood-onset IBD. Our aim was to investigate the body compositions of these patients, with the focus on SMI and fat %.Methods: Body composition was estimated by dual x-ray absorptiometry for 94 young adults with childhood-onset IBD aged 18-27 years, 65 of whom had ulcerative colitis. The Z-scores for SMI, fat %, and BMD were calculated using the normative data from 1,289 individuals with corresponding age. Based on the SMI and fat % Z-scores, each patient was classified as having a body composition profile that was: (i) normal; (ii) obese (fat % Z-score >1); (iii) myopenic (SMI Z-score < -1); or (iv) myopenic-obese.Results: A higher proportion of young adults with childhood-onset IBD had a body composition profile classified as myopenic (24%) or myopenic-obese (9%), as compared to the controls (myopenic [16%, p = .016]; myopenic-obese [2%, p = .002]). Patients with the myopenic or myopenic-obese profile had significantly lower total body BMD Z-scores (-1.3 ± 0.7 and -1.4 ± 0.9, respectively) than patients with the normal profile (-0.2 ± 1.1; p < .001 and p = .004, respectively). Diagnosis of IBD in childhood represented an additional risk for low BMD, regardless of SMI Z-score.Conclusion: Young adults with childhood-onset IBD have a high risk for having altered body composition traits.SummaryYoung adults with childhood-onset IBD carry a high risk for altered body composition traits. The myopenic and myopenic-obese body composition profiles were more frequently observed in patients with IBD than controls, and these profiles were strongly associated with low BMD.
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Composición Corporal/fisiología , Índice de Masa Corporal , Enfermedades Óseas Metabólicas/etiología , Enfermedades Inflamatorias del Intestino/complicaciones , Enfermedades Inflamatorias del Intestino/fisiopatología , Absorciometría de Fotón , Adolescente , Adulto , Densidad Ósea/fisiología , Femenino , Humanos , Modelos Lineales , Estudios Longitudinales , Masculino , Análisis Multivariante , Suecia , Adulto JovenRESUMEN
The gut microbiota harbor a wide range of bacterial species, but also yeasts may be part of this ecosystem. Infants who are being treated in intensive care units are often colonized by Candida species. However, little is known regarding commensal yeast colonization of healthy infants and young children. Here the acquisition of yeast species was studied in a birth-cohort including 133 healthy Swedish infants. A rectal swab sample was obtained on day 3 of life, and fresh fecal samples were obtained at regular intervals up to 3 years of age; the samples were cultured quantitatively for yeasts. Colonization with yeasts increased rapidly in the first months of life, with 73/133 infants (55%) colonized at 6 months of age. The yeast numbers in positive samples decreased from an average of 105 cfu/g in infants aged 0-2 months to 103.5 cfu/g at 3 years of age. Candida albicans was the most frequently isolated species and reached higher population counts than the other species in culture-positive infants. The yeast colonization rate did not differ between infants who were delivered vaginally and those birthed via Caesarean section, whereas breastfed infants showed a lower colonization rate (p < 0.05 for 1 year of age compared to the other infants). The results demonstrate that yeasts, particularly C. albicans and C. parapsilosis (sensu lato), are common commensals in the gut microbiota of healthy infants and young children.
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Candida/fisiología , Candidiasis/microbiología , Portador Sano/microbiología , Heces/microbiología , Microbioma Gastrointestinal , Simbiosis , Candida/clasificación , Candida/aislamiento & purificación , Candida albicans/aislamiento & purificación , Cesárea , Preescolar , Parto Obstétrico , Femenino , Voluntarios Sanos , Humanos , Lactante , Recién Nacido , Estudios Longitudinales , Masculino , SueciaRESUMEN
BACKGROUND: Allergy and other immune-mediated diseases are more frequently reported in children who have undergone liver transplantation. Furthermore, autoantibodies are also prevalent, suggesting a state of immune dysregulation in these patients. Whether or not these processes occur simultaneously in the same individual has not been studied previously. METHODS: A cohort of 43 children who had undergone liver transplantation for nonautoimmune liver disease at median age of 1.3 years was investigated for allergy and autoimmune disease. Sensitization to food and inhalant allergens was assessed, and autoantibodies were measured. RESULTS: The prevalence of food allergy was 26% and that of respiratory allergy was 23%, whereas 33% and 26% of the subjects were sensitized to food and inhalant allergens, respectively. Autoimmune disease (ie, autoimmune hepatitis) occurred in a single individual (2%), whereas autoantibodies were present in 44% of the children. Food allergy and autoantibodies occurred concomitantly in 19% of the children, which was almost twice the frequency expected by chance (11%, P = 0.04). Respiratory allergy and the presence of autoantibodies were unrelated (12% concurrence versus the expected 10%, P = 0.73). In the logistic regression analysis, autoantibody formation was associated with discontinued immunosuppression and food allergy, with odds ratios of 13 (P = 0.01) and 7.1 (P = 0.03), respectively. CONCLUSIONS: In contrast to respiratory allergy, food allergy and autoantibody formation occurred together in the same children who underwent liver transplantation at a frequency higher than would be expected by chance. This may reflect an underlying immune dysregulation that impairs immune tolerance to both food allergens and autoantigens.
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Autoanticuerpos/inmunología , Atresia Biliar/cirugía , Enfermedad Hepática en Estado Terminal/cirugía , Hipersensibilidad a los Alimentos/complicaciones , Trasplante de Hígado , Adolescente , Alérgenos/inmunología , Autoantígenos/inmunología , Enfermedades Autoinmunes/complicaciones , Enfermedades Autoinmunes/inmunología , Atresia Biliar/mortalidad , Niño , Preescolar , Estudios Transversales , Enfermedad Hepática en Estado Terminal/complicaciones , Enfermedad Hepática en Estado Terminal/inmunología , Femenino , Estudios de Seguimiento , Humanos , Terapia de Inmunosupresión , Lactante , Masculino , Oportunidad Relativa , Complicaciones Posoperatorias , Prevalencia , Tacrolimus/uso terapéuticoRESUMEN
OBJECTIVES: Several studies have indicated that early pet keeping could protect the infant from later allergy development. Here, we investigate if there is a dose-dependent association between cat- and dog-keeping during the first year of life and subsequent allergy development. METHODS: Two cohorts were investigated: a cross-sectional questionnaire-based study of 7- to 8-year-old children (N = 1029) from Mölndal and Kiruna, and a birth-cohort of children from the Västra Götaland county clinically evaluated for asthma and allergy by paediatricians up to the age of 8-9 years (N = 249). The cross-sectional study asked validated questions on asthma and allergy that had been used in two previous studies of children from the same areas. In the birth-cohort study, a diagnosis of asthma and allergy was based on predefined clinical criteria, and laboratory evaluation included blood eosinophils, skin-prick tests and specific immunoglobulin E analyses. Information on pets during first year of life was collected retrospectively in the Cross-Sectional Cohort and prospectively in the Birth Cohort. RESULTS: A dose-response association was seen, with less allergic manifestations (any of asthma, allergic rhinoconjunctivitis, or eczema) with increasing number of household cats and dogs during the first year of life. In the Cross-Sectional Cohort, allergy ever decreased from 49% in those with no pets to zero in those with five or more pets (P-value for trend 0.038), and from 32% to zero for allergy last year (P-value for trend 0.006). The same pattern was seen in Birth Cohort. Sensitization to animals, as well as pollens, also decreased with increasing number of animals in the household. CONCLUSION: The prevalence of allergic disease in children aged 7-9 years is reduced in a dose-dependent fashion with the number of household pets living with the child during their first year of life, suggesting a "mini-farm" effect, whereby cats and dogs protect against allergy development.
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Vínculo Humano-Animal , Hipersensibilidad/epidemiología , Hipersensibilidad/prevención & control , Mascotas/fisiología , Alérgenos/farmacología , Animales , Gatos , Niño , Estudios Transversales , Perros , Relación Dosis-Respuesta Inmunológica , Regulación hacia Abajo/inmunología , Femenino , Humanos , Hipersensibilidad/etiología , Hipersensibilidad/inmunología , Masculino , Prevalencia , Estudios Retrospectivos , Factores de Riesgo , Pruebas Cutáneas , Encuestas y CuestionariosRESUMEN
BACKGROUND: Children who have inflammatory bowel disease (IBD) have increased risk of low bone mineral density (BMD). There is a scarcity of information on BMD development through puberty and into young adulthood in patients with childhood-onset IBD. METHODS: We conducted a prospective longitudinal study of BMD in patients with childhood-onset IBD. In total, 74 children with IBD were followed into young adulthood, with a mean follow-up of 8.4 years. The BMD was assessed longitudinally using dual-energy X-ray absorptiometry of the lumbar spine, total hip and whole body, and related to anthropometric measures. RESULTS: Young adult male patients with IBD had lower mean BMD Z-scores for the lumbar spine at -0.8 (±1.1 SD) and total hip at -0.5 (±0.9 SD), as compared to standard references. In young female patients, the BMD Z-scores were within the normal range at all 3 measured sites as compared to the standard references. There were no significant differences in the BMD Z-scores between patients with Crohn's disease and patients with ulcerative colitis. The female and male patients showed significantly improved mean lumbar spine BMD Z-scores during follow-up into young adulthood, indicating that bone accumulation in the lumbar spine continues beyond the expected age for achieving peak bone mass. CONCLUSIONS: Male patients with childhood-onset IBD seem to have an increased risk of compromised BMD in young adulthood. Both female and male patients with IBD seem to increase their BMD beyond the age for expected peak bone mass (see Video abstract, Supplemental Digital Content 1, http://links.lww.com/IBD/B648).
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Densidad Ósea , Enfermedades Óseas Metabólicas/etiología , Enfermedades Inflamatorias del Intestino/complicaciones , Enfermedades Inflamatorias del Intestino/fisiopatología , Absorciometría de Fotón , Adolescente , Adulto , Factores de Edad , Estudios de Casos y Controles , Niño , Femenino , Humanos , Modelos Lineales , Estudios Longitudinales , Masculino , Análisis Multivariante , Estudios Prospectivos , Valores de Referencia , Medición de Riesgo , Factores Sexuales , Adulto JovenRESUMEN
BACKGROUND: Inflammatory bowel disease (IBD) is characterized by gut dysbiosis. To date, the large bowel microbiota has been in focus. However, the microbiota of the small intestine may also be of importance, as the small bowel is a site for the induction and control of mucosal immune responses, which can be modulated by constituents of the local microbiota. METHODS: Duodenal fluids were collected during diagnostic work-up of treatment-naïve children who were suspected of having IBD. The duodenal fluids were analyzed by pyrosequencing (average of 32,000 reads/sample, read length of 500 nucleotides). After diagnosis, the duodenal microbiota of subjects with ulcerative colitis (N = 8) or Crohn's disease (N = 5), and non-IBD controls (N = 8) were compared. RESULTS: Pyrosequencing revealed that the duodenal microbiota of children with ulcerative colitis contained fewer Operational Taxonomic Units (OTUs) per individual than the duodenal microbiota of the controls (P = 0.005). This reduction in richness of the duodenal microbiota was seen for three major phyla: Firmicutes, Actinobacteria, and Bacteroidetes. Several bacterial genera were detected less frequently in the children with ulcerative colitis than in the non-IBD controls, including Collinsella (P = 0.001), Lactobacillus (P = 0.007), and Bacillus (P = 0.007), as well as a non-identified member of the order Sphingobacteriales (P = 0.007). CONCLUSIONS: In this pilot study, we show that the duodenal microbiota of children with ulcerative colitis exhibits reduced overall richness, despite the fact that the inflammation is primarily localized to the colon. These results should be corroborated in a larger study.
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Colitis Ulcerosa/diagnóstico , Duodeno/microbiología , Microbiota , Adolescente , Estudios de Casos y Controles , Niño , Colitis Ulcerosa/microbiología , Femenino , Humanos , Masculino , Proyectos PilotoAsunto(s)
Asma , Ruidos Respiratorios , Asma/diagnóstico , Humanos , Lactante , Estimación de Kaplan-Meier , Medición de Riesgo/métodosRESUMEN
BACKGROUND: Giant papillae tongue disorder (GPTD) is a newly discovered, long-lasting clinical disorder that may develop in organ-transplanted pediatric recipients. The key feature of this disorder is the unique tongue lesion, which comprises swollen fungiform papillae. The aim of this study was to characterize the immunohistopathology of this novel inflammatory condition. METHODS: Six organ transplanted children with GPTD were included in the study. Routine histopathology and immunohistochemical stainings for CD3, CD4, CD8, CD25, FOXP3, CD20, CD138, CD68, CD1a, CD15, CD23, and mast cell tryptase were performed. RESULTS: Immunohistochemical analyses of the oral lesions revealed a subepithelial infiltrate that was primarily composed of CD3- and CD4-positive T cells, CD20-expressing B cells, macrophages, and CD138-positive plasma cells. The CD20-positive cells did not display the typical B cell morphology, having in general a more dendritic cell-like appearance. The CD138-expressing plasma cells were distinctly localized as a dense infiltrate beneath the accumulation of T cells and B cells. Increased numbers of CD1a-expressing Langerhans cells were detected both in the epithelium and connective tissue. Because no granulomas were observed and only single lesional eosinophils were detected, GPTD does not resemble a granulomatous or eosinophilic condition. CONCLUSIONS: We describe for the first time the immunopathological characteristics of a novel inflammatory disorder of the oral cavity, which may develop after solid organ transplantation in children.
Asunto(s)
Inflamación/inmunología , Inflamación/patología , Trasplante de Órganos/efectos adversos , Enfermedades de la Lengua/inmunología , Enfermedades de la Lengua/patología , Adolescente , Adulto , Factores de Edad , Anciano de 80 o más Años , Biomarcadores/análisis , Biopsia , Brasil , Niño , Femenino , Humanos , Inmunohistoquímica , Inmunosupresores/administración & dosificación , Masculino , Persona de Mediana Edad , Suecia , Resultado del TratamientoRESUMEN
BACKGROUND: Although orofacial granulomatosis (OFG) may present as a separate clinical entity, it often seems in conjunction with various systemic diseases, of which Crohn's disease (CD) is one of the most common. The aim of this study was to investigate whether CD with concomitant OFG represents a distinctive disease subtype. METHODS: Twenty-one patients with CD and concomitant OFG (CD+OFG group) were included in the study. As the reference group, a cohort of 39 patients with CD but without OFG (CD-R group) was used. Demographic data and clinical characteristics were recorded at the time of diagnosis. The 2 groups were compared using multivariate analyses. RESULTS: The percentage of patients with intestinal inflammation in the upper gastrointestinal tract was significantly higher in the CD+OFG group, as compared with the CD-R group (81% versus 33%; P < 0.001). Furthermore, ileocolonic inflammation was significantly more common in the CD+OFG patients (81% versus 46%; P = 0.013). In addition, perianal disease was more frequently observed in the CD+OFG group (48% versus 18%; P = 0.033). Significantly more patients showed evidence of granulomas in the primary endoscopy in the CD+OFG group than in the CD-R group (81% versus 38%; P = 0.003). CONCLUSION: The data from this study suggest that the presence of CD in conjunction with OFG represents a distinctive subphenotype of CD that is characterized by extensive inflammation, perianal disease, and pronounced granuloma formation in the intestine.
Asunto(s)
Enfermedad de Crohn/clasificación , Enfermedad de Crohn/complicaciones , Granulomatosis Orofacial/diagnóstico , Adolescente , Adulto , Niño , Femenino , Estudios de Seguimiento , Granulomatosis Orofacial/etiología , Humanos , Masculino , Pronóstico , Adulto JovenRESUMEN
OBJECTIVE: Intestinal and multivisceral transplantation have gained acceptance as treatment modalities for patients with: intestinal failure and life-threatening complications of parenteral nutrition (PN), rare cases of vascular abdominal catastrophes and selected cases of low-grade neoplastic tumors such as neuroendocrine pancreatic tumors and desmoids involving the mesenteric root. The aim was to describe the survival and nutritional outcome in the transplanted Nordic patients and the complications attributed to this procedure. METHOD: The authors included all Nordic patients transplanted between January 1998 and December 2013. Information on patients transplanted outside the Nordic region was collected through questionnaires. RESULTS: A total of 34 patients received different types of intestinal allografts. Currently, there are two Nordic transplant centers (n = 29) performing these procedures (Gothenburg, Sweden n = 24, Helsinki, Finland n = 5). The remaining five patients were transplanted in the USA (n = 3) and the UK (n = 2). Most patients were transplanted for life-threatening failure of PN (70%) caused primarily by intestinal motility diseases (59%). Allograft rejection was the most common complication and occurred in 79% of the patients followed by post-transplantation lymphoproliferative disorders (21%) and graft-versus-host disease (18%). The 1- and 5-year survival was 79% and 65% respectively for the whole cohort and nutritional autonomy was achieved in 73% of the adults and 57% of the children at 1 year after transplantation. CONCLUSION: This collective Nordic experience confirms that intestinal transplantation is a complex procedure with many complications, yet with the possibility to provide long-term survival in selected conditions previously considered untreatable.
Asunto(s)
Rechazo de Injerto/tratamiento farmacológico , Enfermedad Injerto contra Huésped/tratamiento farmacológico , Inmunosupresores/uso terapéutico , Enfermedades Intestinales/terapia , Intestinos/trasplante , Adolescente , Adulto , Anciano , Causas de Muerte , Niño , Preescolar , Femenino , Supervivencia de Injerto , Humanos , Trasplante de Hígado , Masculino , Persona de Mediana Edad , Nutrición Parenteral , Complicaciones Posoperatorias , Países Escandinavos y Nórdicos , Adulto JovenRESUMEN
OBJECTIVES: The aim of this investigation was to characterise and compare the inflammatory infiltrates in patients with orofacial granulomatosis solely (OFG-S) and OFG with coexisting Crohn's disease (OFG+CD). Study DESIGN: Biopsy specimens with granulomas were obtained from patients with OFG-S (n=11) and OFG+CD (n=11) and immunostained with antibodies against CD1a, CD3, CD4, CD8, CD11c, CD20, CD68 and mast cell tryptase, followed by quantitative analysis. RESULTS: Analyses of the connective tissue revealed a significantly higher number of CD3-expressing T cells and CD11c-expressing dendritic cells in the connective tissue of patients with OFG-S compared to patients with OFG+CD. Mast cells displayed a high level of activation, although no significant difference was detected when comparing the two groups. CONCLUSIONS: The results show a different composition of the inflammatory infiltrate in patients with OFG-S compared to patients with OFG+CD. The present observations support that partlydivergent immune mechanisms are involved in these two different subcategories of OFG
Asunto(s)
Humanos , Inmunofenotipificación/métodos , Granulomatosis Orofacial/diagnóstico , Enfermedad de Crohn/epidemiología , Autoinmunidad , Triptasas/análisis , Antígenos CD/análisis , Células DendríticasRESUMEN
OBJECTIVES: The aim of this investigation was to characterise and compare the inflammatory infiltrates in patients with orofacial granulomatosis solely (OFG-S) and OFG with coexisting Crohn's disease (OFG+CD). STUDY DESIGN: Biopsy specimens with granulomas were obtained from patients with OFG-S (n=11) and OFG+CD (n=11) and immunostained with antibodies against CD1a, CD3, CD4, CD8, CD11c, CD20, CD68 and mast cell tryptase, followed by quantitative analysis. RESULTS: Analyses of the connective tissue revealed a significantly higher number of CD3-expressing T cells and CD11c-expressing dendritic cells in the connective tissue of patients with OFG-S compared to patients with OFG+CD. Mast cells displayed a high level of activation, although no significant difference was detected when comparing the two groups. CONCLUSIONS: The results show a different composition of the inflammatory infiltrate in patients with OFG-S compared to patients with OFG+CD. The present observations support that partly-divergent immune mechanisms are involved in these two different subcategories of OFG.