Poster Presentation ESA-SRB 2023 in conjunction with ENSA

Clinical Case of Eosinophilic Esophagitis (EoE) leading to likely diagnosis of congenital Connective Tissue Disease (CTD) (#349)

Dragon Ka Lung Wat 1 , Ingrid Elisabeth Bonapart 2
  1. Resident, MAPU, Toowoomba Hospital, Toowoomba, QLD, Australia
  2. Clinical Director MAPU / Endocrinologist, Toowoomba Hospital, Toowoomba, QLD, Australia

Aims

To raise awareness of the association of EoE with CTD. Risk of EoE is up to 8-fold increase in CTD population(1). Recognizing undiagnosed CTD is of the utmost importance for more favorable outcomes. There is also a role for closer follow up of EoE disease in CTD patients, because of greater risks for more diffused eosinophilic extra-esophageal gastrointestinal disease and resistance to treatments(2).

 

Methods

Patient is a 19M and had 5 admissions for food bolus in 8 years. All of them required gastroscopy to resolve or assess the esophagus. Patient had clinical presentation related to Marfan Syndrome (MFS) and Ehlers Danlos Syndrome (EDS) like tall stature, high-arched palate, pectus incavatum and extensive striae over body. But without typical presentation like long extremities, heart murmur, ectopia lentis, vision issues and flat feet (pes planus) in MFS or hypermobility of joints and hyperflexibility of skin in EDS.

 

Results

Patient had been investigated for MFS at younger age but results were negative. Echocardiogram in 2020 was normal. All 5 gastroscopies were normal, food bolus passed or removed, biopsies had been taken in all gastroscopies and histology from all biopsies showed traditional EoE pictures. 4 out of 5 esophageal biopsies confirmed EoE with the highest count of 300 eosinophils/hpf. He also had positive family history of a sister with confirmed EDS.

 

Conclusion

Patient has been called back for further genetic testing for congenital CTD and screening for different features of CTD with pathology and imaging. We suspect patient to have congenital CTD with features from a few CTD variants. We need to confirm the genetic diagnosis and prevent unfavourable preventable outcomes. Early multi-disciplinary involvement is essential to control symptoms and minimise systemic involvement.

  1. (1) Abonia JP, Wen T, Stucke EM, et al. High prevalence of eosinophilic esophagitis in patients with inherited connective tissue disorders. J Allergy Clin Immunol. 2013;132(2):378-386. doi:10.1016/j.jaci.2013.02.030
  2. (2) Xue Z, Miller TL, Abramson L, et al. Association of eosinophilic esophagitis with autoimmune and connective tissue disorders, and the impact on treatment response. Dis Esophagus. 2022;36(1):doac043. doi:10.1093/dote/doac043