Specific Autoantibodies to Gut Epithelium in Two Infants with Severe Protracted Diarrhoea

Mo, Savage; Mirakian, Rita; Er, Wozniak; Hr, Jenkins; Malone, Margaret; Ad, Phillips; Pj, Milla; Gf, Bottazzo; Jt, Harries

doi:10.1097/00005176-198504000-00006

Cited by 58 publications

(40 citation statements)

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“…Since then, several additional cases of autoimmune enteropathy have been reported (25)(26)(27)(28). Although we could not finely make the diagnosis of autoimmune enteropathy because of the lack of diagnostic modalities including gut epithelial cell autoantibodies (6,25,(28)(29)(30)(31)(32), at least some of the patients in this study might have had autoimmune enteropathy because all cases in the epithelial dysplasia group were unresponsive to elemental milk, dependent on PN, and had considerable villous atrophy. Epithelial dysplasia group included patients with severe intractable diarrhea with normal to minimal villous atrophy with epithelial dysplasia.…”

Section: Discussionmentioning

confidence: 80%

Protracted Diarrhea: Results of the Five-year Survey in a Tertiary Hospital in Korea

2001

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The syndrome of protracted diarrhea (PD) includes several diseases with diverse etiologies. This study was conducted to characterize the spectrum of causes, clinical manifestations, and the outcomes of PD. A retrospective analysis of the clinical and pathological findings was performed on 25 patients with diarrhea starting within the first 2 yr of life and a requirement of parenteral nutrition (PN). According to the intestinal histopathology, patients were classified into four groups: immune enteropathy (12 cases), lymphangiectasia (6 cases), epithelial dysplasia (5 cases), and unclassified (2 cases). All patients with epithelial dysplasia had earlier onset of diarrhea and longer duration of PN than those in the other groups. Three patients (12%) had an evidence of a familial condition. Five patients (three with microvillous inclusion disease and two with immune enteropathy) died. Sixteen patients recovered, and three (two with primary lymphangiectasia and one with microvillous inclusion disease) still had diarrhea. One patient underwent intestinal transplantation for tufting enteropathy. In conclusion, infants with PD should be referred to specialized centers where advanced diagnostic and therapeutic facilities are available, because histological analysis is critical for the diagnosis of PD, and PN or intestinal transplantation is the only therapeutic option in a subset of cases.

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Section: Discussionmentioning

confidence: 80%

Protracted Diarrhea: Results of the Five-year Survey in a Tertiary Hospital in Korea

2001

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“…[11][12][13][14] Increased IELs do occur in a subset of patients with autoimmune enteropathy, which typically appears as a destructive acute inflammatory infiltrate with villous loss that may accompany the absence of goblet and/or Paneth cells. 10,15 Another novel finding in our series is the relative abundance of cases diagnosed as lymphocytic colitis.…”

Section: Discussionmentioning

confidence: 99%

Clinical significance of colonic intraepithelial lymphocytosis in a pediatric population

et al. 2009

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“…Acquired, autoimmune hypothyroidism has rarely been reported before age 3 years [7,8,10] and can be part of a constellation of polyglandular autoimmune endocrine deficiency syndromes [11,12]. We suspect that hypothyroidism was acquired in this case because the results of neonatal thyroid screening were normal on two occasions; thyroid technetium scan revealed a normal appearing gland in its normal location; and anti-thyroid antibodies were significantly elevated.…”

Section: Discussionmentioning

confidence: 78%

Onset of acquired autoimmune hypothyroidism in infancy: a presentation of delayed gross-motor development and rhabdomyolysis

et al. 2006

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We report the case of a 23-month-old girl who presented with poor growth and delayed attainment of gross-motor milestones. Elevated creatine phosphokinase (CPK) indicated rhabdomyolysis, ultimately attributed to severe, acquired autoimmune hypothyroidism. Growth data and bone-age suggest the onset of hypothyroidism occurred at or before 12 months of age. Acquired hypothyroidism is rare before age 3 years, and rhabdomyolysis due to hypothyroidism has not previously been reported as a cause of delayed gross-motor development in toddlerhood. Despite the early onset of hypothyroidism, cognitive function appeared to be unaffected. Adequate thyroid hormone replacement quickly normalized the CPK in our patient, and gross motor development rapidly improved. Although rare, rhabdomyolysis secondary to hypothyroidism should be in the differential diagnosis of delayed gross-motor development in infancy and toddlerhood.

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Specific Autoantibodies to Gut Epithelium in Two Infants with Severe Protracted Diarrhoea

Cited by 58 publications

References 0 publications

Protracted Diarrhea: Results of the Five-year Survey in a Tertiary Hospital in Korea

Protracted Diarrhea: Results of the Five-year Survey in a Tertiary Hospital in Korea

Clinical significance of colonic intraepithelial lymphocytosis in a pediatric population

Onset of acquired autoimmune hypothyroidism in infancy: a presentation of delayed gross-motor development and rhabdomyolysis

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