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1.
Lasers Surg Med ; 51(5): 452-458, 2019 Jul.
Artigo em Inglês | MEDLINE | ID: mdl-30614021

RESUMO

BACKGROUND AND OBJECTIVE: The tethered spectrally-encoded confocal endomicroscopy (SECM) capsule is an imaging device that once swallowed by an unsedated patient can visualize cellular morphologic changes associated with gastrointestinal (GI) tract diseases in vivo. Recently, we demonstrated a tethered SECM capsule for counting esophageal eosinophils in patients with eosinophilic esophagitis (EoE) in vivo. Yet, the current tethered SECM capsule is far too long to be widely utilized for imaging pediatric patients, who constitute a major portion of the EoE patient population. In this paper, we present a new tethered SECM capsule that is 33% shorter, has an easier and repeatable fabrication process, and produces images with reduced speckle noise. MATERIALS AND METHODS: The smaller SECM capsule utilized a miniature condenser to increase the fiber numerical aperture and reduce the capsule length. A custom 3D-printed holder was developed to enable easy and repeatable device fabrication. A dual-clad fiber (DCF) was used to reduce speckle noise. RESULTS: The fabricated SECM capsule (length = 20 mm; diameter = 7 mm) had a similar size and shape to a pediatric dietary supplement pill. The new capsule achieved optical sectioning thickness of 13.2 µm with a small performance variation between devices of 1.7 µm. Confocal images of human esophagus obtained in vivo showed the capability of this new device to clearly resolve microstructural epithelial details with reduced speckle noise. CONCLUSIONS: We expect that the smaller size and better image performance of this new SECM capsule will greatly facilitate the clinical adoption of this technology in pediatric patients and will enable more accurate assessment of EoE-suspected tissues. Lasers Surg. Med. 51:452-458, 2019. © 2019 Wiley Periodicals, Inc.

2.
Pediatr Int ; 57(5): 1028-30, 2015 Oct.
Artigo em Inglês | MEDLINE | ID: mdl-26508189

RESUMO

Prior studies have reported disparate clinical presentations between children with celiac disease and type 1 diabetes mellitus and those with celiac disease alone. Studies focusing on differences in endoscopic and histopathological findings, however, are limited. We reviewed children aged 2-18 years, presenting for an initial evaluation between January 2000 and December 2010. Data on medical history, serologic markers, upper endoscopy, and histopathology were collected. Only the children with positive celiac serology who had upper endoscopy performed within 3 months of the initial visit were included. We identified 294 children who fulfilled the criteria, 21 of whom had diagnosed type 1 diabetes mellitus. Diabetic children were more likely to have absence of gastrointestinal symptoms. Erythematous duodenal and esophageal mucosa on endoscopy, and histopathology suggestive of reflux esophagitis were more common in the diabetes group. Diabetic children with positive celiac serology had different histopathological features as compared with their non-diabetic counterparts.


Assuntos
Doença Celíaca/complicações , Diabetes Mellitus Tipo 1/complicações , Mucosa Intestinal/imunologia , Adolescente , Biópsia , Doença Celíaca/diagnóstico , Doença Celíaca/imunologia , Criança , Pré-Escolar , Diabetes Mellitus Tipo 1/diagnóstico , Feminino , Seguimentos , Gastroscopia , Humanos , Mucosa Intestinal/patologia , Masculino , Estudos Retrospectivos
3.
J Pediatr ; 162(3): 501-4, 2013 Mar.
Artigo em Inglês | MEDLINE | ID: mdl-23084709

RESUMO

OBJECTIVE: To investigate whether season of birth is associated with celiac disease (CD). STUDY DESIGN: We performed a medical record review of 1964 patients with biopsy-proven CD at 3 teaching hospitals (2 pediatric centers and 1 adult center) between 2000 and 2010. The first positive small intestinal biopsy result defined age of diagnosis. The observed proportions of births in each season (spring [March-May], summer [June-August], fall [September-November], and winter [December-February]) were compared with the expected proportions using binomial probability tests. RESULTS: The mean age at diagnosis was 9.8 ± 5.0 years in the 2 pediatric centers and 43.6 ± 15.8 years in the adult center. The cohort was predominately female (69%). Overall, more patients were born in spring (27%) than in any other season: summer (25%), fall (25%), and winter (23%). In patients diagnosed before age 15 years, the spring birth excess was present in boys (33%; P = .0005), but not in girls (26%; P = .43). The sex difference in season of birth was less striking in patients with CD diagnosed at age ≥15 years. CONCLUSION: Season of birth is an environmental risk factor for CD, particularly in boys diagnosed before age 15 years. The results are consistent with a new theoretical model that integrates potential environmental factors (eg, gluten introduction, ultraviolet-B exposure, vitamin D status) and acute viral gastrointestinal infections in early childhood.


Assuntos
Doença Celíaca/epidemiologia , Parto , Adolescente , Adulto , Idoso , Idoso de 80 Anos ou mais , Criança , Pré-Escolar , Feminino , Humanos , Lactente , Masculino , Pessoa de Meia-Idade , Modelos Biológicos , Fatores de Risco , Estações do Ano , Adulto Jovem
4.
J Pediatr ; 161(3): 471-5, 2012 Sep.
Artigo em Inglês | MEDLINE | ID: mdl-22484356

RESUMO

OBJECTIVES: To determine independent predictors of gluten avoidance and of a physician's decision to initiate a gluten-free diet (GFD) in children and adolescents without confirmed celiac disease. STUDY DESIGN: We performed a structured medical record review of 579 patients aged 1-19 years presenting for evaluation of celiac disease between January 2000 and December 2010 at a large Boston teaching hospital. We collected data including demographic information, medical history, serology, small intestinal biopsy, history of gluten avoidance, and the postworkup recommendation of implementation of a GFD. Predictors of gluten-related issues were identified by multivariate logistic regression. RESULTS: Among 579 children without a previous diagnosis of celiac disease (mean age, 8.7 years), 43 (7.4%) had ever avoided gluten. Independent predictors of gluten avoidance were irritability or poor temper (OR, 3.2), diarrhea (OR, 2.5), weight issues (OR, 0.4), pervasive developmental disorder (OR, 5.3), and family history of celiac disease (OR, 2.2). Among 143 children without confirmed celiac disease who underwent diagnostic evaluation, several predictive factors were associated with a physician- recommended/parent-initiated GFD: irritability (OR, 6.4), diarrhea (OR, 3.4), pervasive developmental disorder (OR, 7.9), and positive serology before the referral (OR, 4.3). CONCLUSION: Gluten avoidance among children and adolescents without a previous diagnosis of celiac disease is relatively common. The identified predictors suggest that gluten avoidance is associated with nonspecific behavioral and gastrointestinal complaints and perhaps with the perceived dietary responses in another family member thought to have celiac disease.


Assuntos
Doença Celíaca/dietoterapia , Dieta Livre de Glúten , Comportamento Alimentar , Adolescente , Criança , Pré-Escolar , Tomada de Decisões , Feminino , Humanos , Modelos Logísticos , Masculino , Padrões de Prática Médica
5.
Gastrointest Endosc ; 74(5): 992-1000, 2011 Nov.
Artigo em Inglês | MEDLINE | ID: mdl-21944314

RESUMO

BACKGROUND: Diagnosis of eosinophilic esophagitis (EoE) currently requires endoscopic biopsy and histopathologic analysis of the biopsy specimens to count intraepithelial eosinophils. Reflectance confocal microscopy (RCM) is an endomicroscopy technology that is capable of obtaining high-resolution, optically sectioned images of esophageal mucosa without the administration of exogenous contrast. OBJECTIVE: In this study, we investigated the capability of a high-speed form of RCM, termed spectrally encoded confocal microscopy (SECM), to count intraepithelial esophageal eosinophils and characterize other microscopic findings of EoE. DESIGN: A total of 43 biopsy samples from 35 pediatric patients and 8 biopsy samples from 8 adult patients undergoing EGD for EoE were imaged by SECM immediately after their removal and then processed for routine histopathology. Two SECM readers, trained on adult cases, prospectively counted intraepithelial eosinophils and detected the presence of abscess, degranulation, and basal cell hyperplasia on SECM images from the pediatric patients. A pathologist blinded to the SECM data analyzed the same from corresponding slides. SETTING: The Gastrointestinal Unit, Massachusetts General Hospital. RESULTS: Eosinophils by SECM demonstrated a higher reflectance than the surrounding cells and other inflammatory cells. There was good correlation between SECM and histology maximum eosinophil counts/high-power field (R = 0.76, P < .0001). Intra- and interobserver correlations for SECM counts were very good (R = 0.93 and R = 0.92, respectively; P < .0001). For the commonly used eosinophil count cutoff of 15 per high-power field, the sensitivity and specificity of SECM for EoE were 100%. The sensitivity and specificity for abscess, degranulation, and basal cell hyperplasia were 100% and 82%, 91% and 60%, and 94% and 80%, respectively. Intra- and interobserver agreements for these microscopic features of EoE were very good (κ = 0.9/0.9, 0.84/1.0, 0.91/0.81, respectively). LIMITATION: Ex vivo study. CONCLUSIONS: This study demonstrates that RCM can be used to accurately count intraepithelial eosinophils and identify other microscopic abnormalities associated with EoE on freshly excised biopsy samples. These findings suggest that RCM may be developed into a tool for assessing eosinophilic infiltration in the esophagus in vivo.


Assuntos
Abscesso/patologia , Degranulação Celular , Esofagite Eosinofílica/patologia , Eosinófilos/fisiologia , Esôfago/patologia , Microscopia Confocal , Adolescente , Adulto , Biópsia , Contagem de Células , Criança , Pré-Escolar , Endoscopia do Sistema Digestório , Eosinófilos/patologia , Feminino , Humanos , Hiperplasia/patologia , Lactente , Masculino , Mucosa/patologia , Variações Dependentes do Observador , Projetos Piloto , Sensibilidade e Especificidade , Adulto Jovem
6.
AJR Am J Roentgenol ; 197(1): 224-31, 2011 Jul.
Artigo em Inglês | MEDLINE | ID: mdl-21701034

RESUMO

OBJECTIVE: The objectives of this study were prospective evaluation of MR enterographic accuracy for detecting Crohn disease imaging features in pediatric patients, compared with a CT reference standard, as well as determination of MR enterographic accuracy for detecting active bowel inflammation and fibrosis using a histologic reference standard. SUBJECTS AND METHODS: The study group for this blinded prospective study included 21 pediatric subjects with known Crohn disease scheduled for clinical CT and histologic bowel sampling for symptomatic exacerbation. All subjects and their parents gave informed consent to also undergo MR enterography. CT and MR enterography examinations were independently reviewed by two radiologists and were scored for Crohn disease features. All bowel histology specimens were reviewed by a single pathologist for the presence of active mucosal inflammation and mural fibrosis, followed by correlation of imaging and histologic findings. RESULTS: All 21 subjects underwent MR enterography and histologic sampling, 18 of whom also underwent CT. MR enterography had high sensitivity for detecting Crohn disease imaging features (e.g., bowel wall thickening, mesenteric inflammation, lymphadenopathy, fistula, and abscess) compared with CT, with individual sensitivity values ranging from 85.1% to 100%. Of a total of 53 abnormal bowel segments with correlation of MRI and histologic findings, MR enterography showed 86.7% accuracy (90.0% sensitivity and 82.6% specificity) for detecting active inflammation (p < 0.001). The accuracy of MR enterography for detecting mural fibrosis overall was 64.9%, compared with histology, but increased to 83.3% (p < 0.05) for detecting fibrosis without superimposed active inflammation. CONCLUSION: MR enterography can substitute for CT as the first-line imaging modality in pediatric patients with Crohn disease, on the basis of its ability to detect intestinal pathologic abnormalities in both small and large bowel as well as extraintestinal disease manifestations. Additionally, MR enterography provides an accurate noninvasive assessment of Crohn disease activity and mural fibrosis and can aid in formulating treatment strategies for symptomatic patients and assessing therapy response.


Assuntos
Doença de Crohn/patologia , Intestinos/patologia , Imageamento por Ressonância Magnética/métodos , Adolescente , Criança , Feminino , Humanos , Masculino , Estudos Prospectivos , Reprodutibilidade dos Testes , Sensibilidade e Especificidade , Adulto Jovem
8.
Sci Rep ; 8(1): 2631, 2018 02 08.
Artigo em Inglês | MEDLINE | ID: mdl-29422678

RESUMO

Esophagogastroduodenoscopy (EGD) is a widely used procedure, posing significant financial burden on both healthcare systems and patients. Moreover, EGD is time consuming, sometimes difficult to tolerate, and suffers from an imperfect diagnostic yield as the limited number of collected biopsies does not represent the whole organ. In this paper, we report on technological and clinical feasibility of a swallowable tethered endomicroscopy capsule, which is administered without sedation, to image large regions of esophageal and gastric mucosa at the cellular level. To demonstrate imaging capabilities, we conducted a human pilot study (n = 17) on Eosinophilic Esophagitis (EoE) patients and healthy volunteers from which representative cases are presented and discussed. Results indicate that, compared to endoscopic biopsy, unsedated tethered capsule endomicroscopy obtains orders of magnitude more cellular information while successfully resolving characteristic tissue microscopic features such as stratified squamous epithelium, lamina propria papillae, intraepithelial eosinophils, and gastric cardia and body/fundic mucosa epithelia. Based on the major import of whole organ, cellular-level microscopy to obviate sampling error and the clear cost and convenience advantages of unsedated procedure, we believe that this tool has the potential to become a simpler and more effective device for diagnosing and monitoring the therapeutic response of EoE and other esophageal diseases.


Assuntos
Esofagite Eosinofílica/diagnóstico , Esôfago/ultraestrutura , Mucosa Gástrica/ultraestrutura , Microscopia Confocal/métodos , Adulto , Feminino , Humanos , Masculino , Projetos Piloto
9.
J Pediatr Gastroenterol Nutr ; 44(3): 312-7, 2007 Mar.
Artigo em Inglês | MEDLINE | ID: mdl-17325550

RESUMO

OBJECTIVES: We evaluated the response to infliximab in pediatric patients with ulcerative colitis (UC) and their long-term follow-up. We expanded our previous study of 14 patients and furthermore evaluated the success of weaning patients from infliximab. PATIENTS AND METHODS: We reviewed the charts of 27 pediatric patients with UC who were treated with infliximab instead of undergoing a colectomy. Patients with new-onset UC refractory to intravenous steroids for 5 to 10 days and patients with non-steroid-dependent UC with an acute exacerbation were classified as acutely ill (n = 16); patients with chronic steroid-dependent UC were classified as chronically ill (n = 11). The Lichtiger Colitis Activity Index (LCAI) was measured for all patients at baseline and at 1 and 2 months after treatment with infliximab was initiated. Patients were regarded as successfully treated if they remained off steroids and avoided colectomy. RESULTS: The acutely ill group had a mean LCAI score of 11.4 at induction and 0.3 after 2 months. The chronically ill group had a mean LCAI score of 11.2 at induction and 5.5 after 2 months. Treatment with infliximab was successful in 75% of acutely ill patients and in 27% of chronically ill patients. Infliximab was discontinued in 80% of successfully treated patients (83% of acutely ill, 67% of chronically ill). These patients had an average of 10 infusions and a mean follow-up time of 10 months from their last infliximab infusion. CONCLUSIONS: Our results suggest that infliximab is more effective in acutely ill UC patients than in patients with chronic steroid-dependent UC. In addition, some patients treated with infliximab can be weaned from infliximab and maintain remission.


Assuntos
Anti-Inflamatórios/uso terapêutico , Anticorpos Monoclonais/uso terapêutico , Colite Ulcerativa/tratamento farmacológico , Adolescente , Adulto , Criança , Pré-Escolar , Feminino , Seguimentos , Humanos , Fatores Imunológicos/uso terapêutico , Infliximab , Masculino , Fatores de Tempo
10.
Nutrition ; 31(1): 236-8, 2015 Jan.
Artigo em Inglês | MEDLINE | ID: mdl-25441597

RESUMO

OBJECTIVE: Prior studies have shown that dietary gluten avoidance (DGA) is relatively common in children without previously diagnosed celiac disease (CD), and several clinical predictors of DGA have been found. However, available data on predictors of DGA in adults without diagnosed CD are limited. The aim of this study was to determine the independent predictors of DGA in this population. METHODS: We performed a structured medical record review of 376 patients, ages ≥ 20 y, who had never been formally diagnosed with CD, presenting for an initial CD evaluation (ICD-9-CM 579.0) between January 2000 and December 2010 at two large Boston teaching hospitals. We collected data including demographic characteristics, medical history, history of CD serology before referral, and self-reported DGA. Predictors of DGA were determined using multivariable logistic regression. RESULTS: Mean age was 47 (SD = 17) years. We found that 41 patients (10.9%; 95% confidence interval [CI], 7.9-14.5) had avoided gluten at some time in their lives. Most patients had subjective abdominal complaints or bowel movement changes. History of CD seropositivity before referral was noted in 14%. Independent predictors of DGA (P < 0.05) were lactose intolerance (odds ratio [OR], 2.8; 95% CI, 1.1-7.5), food allergy (OR, 3.8; 95% CI, 1.04-13.7), and history of positive serology of less-specific CD markers before the referral (OR, 3.2; 95% CI, 1.3-7.9). CONCLUSIONS: Gluten avoidance is common in a clinic population of adults without prior CD diagnosis. The recognized predictors suggest that DGA may associate with conditions presenting with nonspecific gastrointestinal complaints and perhaps with the perceived benefits of DGA among patients with prior history of positive CD serology.


Assuntos
Doença Celíaca/epidemiologia , Dieta Livre de Glúten , Adulto , Biomarcadores/sangue , Doença Celíaca/diagnóstico , Feminino , Gastroenteropatias/diagnóstico , Gastroenteropatias/epidemiologia , Glutens/administração & dosagem , Humanos , Modelos Logísticos , Masculino , Pessoa de Meia-Idade , Análise Multivariada , Adulto Jovem
11.
J Allergy Clin Immunol Pract ; 3(6): 951-6, 2015.
Artigo em Inglês | MEDLINE | ID: mdl-26342740

RESUMO

BACKGROUND: One half to one third of the patients with eosinophilic esophagitis (EoE) do not achieve histological remission on initial treatment. We wondered whether these treatment failure patients are a distinct clinical subset. OBJECTIVE: To analyze EoE treatment outcomes in a predominantly pediatric population. METHODS: We reviewed 100 serial EoE cases at Massachusetts General Hospital starting from 2007. We defined histological remission as peak esophageal eosinophil count of less than 10/hpf. RESULTS: Ninety-seven patients with EoE underwent initial treatments: 54 of 81 (67%) responded to dietary therapy, and 9 of 16 (56%) responded to topical glucocorticoids. Of the 34 who failed initial treatment, 24 underwent various second treatment regimens and 54% (13 of 24) responded. Eight of the remaining 11 who failed second treatment underwent additional treatments and 2 ultimately responded. The overall response rate by intent-to-treat analysis increased from 65% (63 of 97) with initial treatment to 78% (76 of 97) with rescue treatment, and further to 80% (78 of 97) with multiple treatments. On a per-protocol basis, the overall response rate was 93% (78 of 84); however, patients who failed the first 2 rounds of therapy had only a 20% response rate. Patients who responded to initial treatment were found to have more symptoms and endoscopic abnormalities. Comparison of patients who failed both initial and rescue therapy with those who responded to rescue therapy did not identify any differentiating clinical features. CONCLUSIONS: More than half of the patients who failed initial EoE treatment could still achieve histological remission with individualized rescue treatments. No clinical features could predict response to rescue treatment.


Assuntos
Esofagite Eosinofílica/dietoterapia , Esofagite Eosinofílica/tratamento farmacológico , Adolescente , Antialérgicos/uso terapêutico , Antiasmáticos/uso terapêutico , Budesonida/uso terapêutico , Criança , Estudos de Coortes , Dieta/métodos , Endoscopia do Sistema Digestório , Esofagite Eosinofílica/imunologia , Feminino , Fluticasona/uso terapêutico , Seguimentos , Glucocorticoides/uso terapêutico , Humanos , Masculino , Estudos Retrospectivos , Falha de Tratamento , Resultado do Tratamento
12.
Inflamm Bowel Dis ; 10(4): 417-20, 2004 Jul.
Artigo em Inglês | MEDLINE | ID: mdl-15475750

RESUMO

OBJECTIVE: We describe the prolonged clinical benefit of murine chimeric antitumor necrosis factor (TNF)-alpha monoclonal antibody, infliximab, on pediatric patients with Crohn's disease and ileal pouch anal anastomosis (IPAA). METHODS: A retrospective review of patients originally diagnosed with ulcerative colitis, status post colectomy and IPAA, who developed findings compatible with Crohn's disease was undertaken. Refractory pouchitis developed in all patients as well as protracted symptoms of diarrhea, abdominal pain, joint pain, and incontinence. All patients received infliximab. RESULTS: Four pediatric patients (2 males and 2 females) with mean age of 14.5 years (range 11-18 years) were studied. The development of perianal fistulas in 2 patients, granuloma on biopsy in 1 patient and perianal skin tag in 1 patient, led to a diagnosis change of CD. After failure to respond to antibiotics, aminosalicylates and immunomodulators such as azathioprine and 6-mercaptopurine (6-MP), all patients were treated with infliximab. Patients received infliximab infusions at a dose of 5 mg/kg, initially at weeks 0, 2 and 6 and subsequently at 8 weeks intervals in combination with an immunomodulator drug. All patients showed marked improvement clinically, endoscopically, and histologically. CONCLUSION: Infliximab can be used successfully for the treatment of pediatric patients with Crohn's disease and IPAA who are refractory to conventional therapies.


Assuntos
Anticorpos Monoclonais/uso terapêutico , Colite Ulcerativa/cirurgia , Fármacos Gastrointestinais/uso terapêutico , Pouchite/tratamento farmacológico , Proctocolectomia Restauradora/efeitos adversos , Adolescente , Canal Anal/cirurgia , Anticorpos Monoclonais/farmacologia , Criança , Feminino , Fármacos Gastrointestinais/farmacologia , Humanos , Infliximab , Masculino , Estudos Retrospectivos , Resultado do Tratamento , Fator de Necrose Tumoral alfa
13.
APMIS ; 121(4): 266-71, 2013 Apr.
Artigo em Inglês | MEDLINE | ID: mdl-23030455

RESUMO

Positive coeliac serology with normal villous morphology (NVM) indicates potential coeliac disease (CD). Few studies have compared characteristics of NVM vs villous atrophy in patients with positive serology. Our aim was to determine the independent clinical predictors of NVM in children with positive CD serology. We performed a structured medical record review of patients aged 1-19 years who presented for an initial CD evaluation between 2000 and 2010 at a large teaching hospital. Data collection included demographics, medical history, prior history of gluten avoidance, CD-specific serology, oesophagogastroduodenoscopy and histopathology. Predictors of NVM (vs Marsh III) were determined using multivariable logistic regression. Among 320 patients with positive serology, we identified 62 patients (19%, 95% CI 15-24) with NVM (i.e. potential CD). Younger children may have been more likely to exhibit NVM (p = 0.06). Three significant predictors of NVM were prior gluten avoidance (OR 4.17, 95% CI 1.02-17.13), positive tissue transglutaminase antibody but <100 U/mL (OR 14.75, 95% CI 3.33-65.30), and absence of gross duodenal abnormalities (OR 3.48, 95% CI 1.51-8.03). Among children with positive CD serology, prior gluten avoidance predicts NVM. Future studies are warranted on the impact of gluten intake and CD testing in children without prior established CD diagnosis.


Assuntos
Anticorpos/sangue , Doença Celíaca/patologia , Proteínas de Ligação ao GTP/imunologia , Transglutaminases/imunologia , Adolescente , Adulto , Doença Celíaca/imunologia , Criança , Pré-Escolar , Feminino , Humanos , Lactente , Modelos Logísticos , Masculino , Proteína 2 Glutamina gama-Glutamiltransferase
14.
Ann Epidemiol ; 23(3): 136-42, 2013 Mar.
Artigo em Inglês | MEDLINE | ID: mdl-23313264

RESUMO

PURPOSE: To evaluate the value of ICD-9-CM code for identifying celiac disease (CD). METHODS: We searched administrative data to identify all adults with ICD-9-CM diagnosis code 579.0 (CD) at three teaching hospitals between 2000 and 2010. We then stratified patients according to the presence/absence of relevant serology and endoscopy codes into four groups: None, serology, endoscopy, and both. A diagnostic algorithm was applied to define CD status. RESULTS: Through random sampling and appropriate weighting, the 1200 reviewed patients represented a cohort of 8,122 cases. The overall positive predictive value (PPV) of the ICD-9-CM code was 15% (95% confidence interval [CI], 13%-17%). Case identification by a diagnosis code alone had a PPV of 4%, whereas the group with diagnosis code plus both serology and endoscopy testing had a PPV of 49%. Independent predictors of CD were non-Hispanic white, ICD-9-CM-coded patient group, total number of a diagnosis code, and receiving a diagnosis code by a gastroenterologist. The model had an area under the curve of 0.87 (95% CI, 0.84-0.89). CONCLUSIONS: The performance of ICD-9-CM 579.0 alone for identifying CD is extremely poor. Adding other readily available administrative data significantly improves CD case identification. The proposed case finding strategy via administrative databases may facilitate future research on CD.


Assuntos
Algoritmos , Doença Celíaca/diagnóstico , Doença Celíaca/epidemiologia , Bases de Dados Factuais/estatística & dados numéricos , Classificação Internacional de Doenças , Adulto , Idoso , Idoso de 80 Anos ou mais , Doença Celíaca/classificação , Estudos de Coortes , Feminino , Humanos , Masculino , Massachusetts/epidemiologia , Pessoa de Meia-Idade , Valor Preditivo dos Testes , Prevalência
16.
Clin Transl Gastroenterol ; 3: e9, 2012 Feb 16.
Artigo em Inglês | MEDLINE | ID: mdl-23238134

RESUMO

OBJECTIVES: Celiac disease (CD) is common and often cited as an "iceberg" phenomenon (i.e., an assumed large number of undiagnosed cases). Recently, atypical or asymptomatic manifestations are becoming more commonly described in older children and adolescents. Moreover, CD diagnosis in children can be complicated by several factors, including its diverse clinical presentations, delay in recognizing CD signs and symptoms, and premature dietary gluten avoidance before the formal diagnosis of CD. To date, few studies have directly examined age-related differences in clinical characteristics and gluten-related issues among children with CD. The aim of this study was to determine age-related patterns in clinical characteristics and gluten-related issues among children with confirmed CD. METHODS: We performed a structured medical record review of biopsy-proven CD patients, aged 0-19 years, between 2000 and 2010 at a large Boston teaching hospital. Data collection included demographics, medical history, gluten-related issues, and diagnostic investigations (CD-specific serology, upper gastrointestinal endoscopy, and small intestinal biopsy). The first positive duodenal biopsy with Marsh III classification defined age of diagnosis. Patients were divided into three age groups for comparisons of the aforementioned characteristics: infant-preschool group (0-5 years), school-aged group (6-11 years), and adolescence group (12-19 years). RESULTS: Among 411 children with biopsy-proven CD, the mean age was 9.5 (s.d. 5.1) years. Most were female (63%) and white (96%). All children had positive CD-specific serology. Most children presented with either abdominal complaints or bowel movement changes. Overall, boys were more common among infant-preschool group compared with the other age groups. More distinct clinical manifestations (vomiting, bowel movement changes, and weight issues) were apparent in the youngest group, whereas school-aged children had more subjective abdominal complaints at the initial presentation. Conversely, the adolescents were most likely to present without any gastrointestinal (GI) symptoms, but not when this was combined with absence of weight issues. Age of diagnosis was not associated with atypical extraintestinal CD presentations. Regarding the gluten-related issues, 10% of school-aged children avoided dietary gluten before the formal CD diagnosis, and 27% of the adolescents reported dietary gluten transgression within the first 12 months of diagnosis, significantly higher than the other age groups. Age differences in histopathology were also found. Whereas the infant-preschool group had a higher proportion of total villous atrophy, the older children were more likely to have gross duodenal abnormalities and chronic duodenitis suggestive of CD at the time of diagnosis. CONCLUSIONS: Children and adolescents with CD have age-related patterns in both the clinical presentations and gluten-related issues. More pronounced clinical and histological features were determined in younger children, whereas older children more commonly presented with solely subjective abdominal complaints or even without any GI symptoms. However, silent and atypical extraintestinal CD presentations were comparable between age groups. In addition to the aforementioned presentations, the higher rates of dietary gluten avoidance and transgression in older children make CD diagnosis and management particularly challenging. These age-related patterns may further increase awareness, facilitate early diagnosis, and improve patient care of pediatric CD.

17.
J Clin Pathol ; 65(9): 791-4, 2012 Sep.
Artigo em Inglês | MEDLINE | ID: mdl-22718844

RESUMO

AIMS: Studies on the role of duodenal bulb biopsy (DBB) in coeliac disease (CD) evaluation have increased in recent years; growing evidence suggests that the disease can present solely in the duodenal bulb. Moreover, recent CD guidelines recommend obtaining a DBB. The study aim was to examine DBB performance in children undergoing CD evaluation and to identify independent predictors of DBB performance. METHODS: The authors performed a structured chart review of children aged <18 years who underwent CD evaluation between 2000 and 2010 at a large teaching hospital. The authors collected data including demographics, serology, endoscopy and histopathological findings. Predictors of DBB performance (obtained vs not obtained) were determined using multivariable logistic regression. RESULTS: Among 616 children who underwent endoscopy, DBB was performed in 103 children (17%, 95% CI 14% to 20%) with an increasing trend in the more recent years (2008-2010, 25%; 2004-2007, 16%; and 2000-2003, 5%, p<0.001). Three independent predictors of DBB performance were older age at endoscopy (OR 1.05 per year of age), gross gastric antral abnormalities (OR 2.81) and gross duodenal abnormalities (OR 5.55). Regarding the DBB histological findings, patchiness of CD was found in 15%. Positive Marsh III biopsy presented solely on the DBB in 6/103 (6%, 95% CI 2% to 12%) children. CONCLUSIONS: The authors found a significant increase in DBB performance over time, but the overall performance remains suboptimal. Improving education on obtaining a DBB for CD evaluation is crucial, especially among those children in whom DBB is more likely to be omitted.


Assuntos
Doença Celíaca/patologia , Duodeno/patologia , Mucosa Intestinal/patologia , Adolescente , Fatores Etários , Biópsia/tendências , Boston , Distribuição de Qui-Quadrado , Criança , Pré-Escolar , Endoscopia Gastrointestinal/tendências , Feminino , Humanos , Lactente , Recém-Nascido , Modelos Logísticos , Masculino , Análise Multivariada , Razão de Chances , Valor Preditivo dos Testes , Prognóstico , Fatores de Tempo
18.
J Pediatr Gastroenterol Nutr ; 39(2): 166-70, 2004 Aug.
Artigo em Inglês | MEDLINE | ID: mdl-15269622

RESUMO

PURPOSE: The authors report their experience with infliximab in pediatric patients with ulcerative colitis (UC). METHODS: Fourteen patients were reviewed. Group 1 included five patients with newly diagnosed, fulminant colitis refractory to 7 to 10 days of intravenous steroids. Group 2 included four patients with ulcerative colitis in remission off steroid therapy who experienced relapse and were hospitalized with fulminant colitis refractory to intravenous steroids for 7 to 10 days. Group 3 included five patients chronically dependent on steroids with colitis refractory to medical management. All patients were treated on an open-label basis with infliximab infusions of 5 mg/kg/dose at 0, 2, and 6 weeks and every 6 to 8 weeks thereafter. Follow-up was maintained for at least 6 weeks. Clinical status was scored with the Lichtiger Colitis Activity Index (LCAI) at each visit. LCAI >or=10 was considered treatment failure. We defined success as LCAI or=11 before infliximab treatment. All group 1 patients experienced response to infliximab. All but one (75%) patient in group 2 had a response. Only one (20%) group 3 patient had a response to infliximab. CONCLUSION: Infliximab was an effective agent in the treatment of acute UC in our patients. Long-term steroid use and emergency colectomy were avoided. Infliximab was less effective in patients who were dependent on steroids.


Assuntos
Anticorpos Monoclonais/uso terapêutico , Colite Ulcerativa/tratamento farmacológico , Fármacos Gastrointestinais/uso terapêutico , Doença Aguda , Adolescente , Adulto , Criança , Doença Crônica , Feminino , Humanos , Infliximab , Infusões Parenterais , Masculino , Indução de Remissão , Estudos Retrospectivos , Índice de Gravidade de Doença , Resultado do Tratamento
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