Clinical, pathologic, and molecular characterization of familial eosinophilic esophagitis compared with sporadic cases

Clin Gastroenterol Hepatol. 2008 Jun;6(6):621-9. doi: 10.1016/j.cgh.2008.01.004. Epub 2008 Apr 22.

Abstract

Background & aims: Eosinophilic esophagitis (EE) occurs in families.

Methods: Record review confirmed patient kinship and provided clinical information. Slide review confirmed the diagnosis (threshold peak number > or = 24 eosinophils/high-power field).

Results: Fifty-nine members (41 males, 18 females) of 26 families were 3 months to 47 years of age (mean age, 10.3 y) at diagnosis. The only recorded race was Caucasian. In 4 families a parent of an affected male had EE. The most common complaint at diagnosis was dysphagia (68% of patients). Endoscopy showed esophageal mucosal furrows (93% of patients) and exudates (44%). Fifty-one percent had asthma. Skin prick tests to food and aeroallergens were positive in 76% and 71%, respectively. Familial EE characteristics (clinical, endoscopic, pathologic, and global esophageal transcript expression profile analysis) were similar to sporadic EE, except among patients with mucosal furrows: familial patients had lower peak eosinophil counts in the distal esophagus (P = .03) compared with sporadic patients. The basic characteristics of EE (eg, eosinophil levels, rate of atopy) did not vary with patient age. By using genome-wide microarray analysis, no significant differences (P < .05, false-discovery rate) were observed between familial and sporadic EE. Among all patients, chest pain was more common in females (P = .02), and thickened mucosa was more common in males (P = .006).

Conclusions: These data support a familial pattern of inheritance of EE and a pathogenesis shared with sporadic EE. EE should be considered in symptomatic family members of patients who have EE.

Publication types

  • Comparative Study
  • Research Support, N.I.H., Extramural
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adolescent
  • Adult
  • Asthma
  • Child
  • Child, Preschool
  • Deglutition Disorders / etiology
  • Eosinophils / immunology*
  • Esophagitis / genetics
  • Esophagitis / immunology
  • Esophagitis / pathology*
  • Esophagitis / physiopathology*
  • Esophagoscopy
  • Family Health*
  • Female
  • Gene Expression Profiling
  • Humans
  • Infant
  • Male
  • Middle Aged
  • Mucous Membrane / pathology
  • Oligonucleotide Array Sequence Analysis
  • White People