Esophageal squamous papillomas with focal dermal hypoplasia and eosinophilic esophagitis

Abstract

Focal dermal hypoplasia (FDH) is a rare disorder of the mesodermal and ectodermal tissues. Here we present an eight-year-old female known to have FDH who presents with poor weight gain and dysphagia. She was diagnosed with multiple esophageal papillomas and eosinophilic esophagitis. She was successfully treated with argon plasma coagulation and ingested fluticasone propionate, which has not been described previously in a child.

A Rare Case of Squamous Cell Carcinoma of the Esophagus in a Patient With Goltz Syndrome

“…20,21 Per literature review, there are only 5 cases of esophageal papillomas associated with Goltz syndrome. 12,[22][23][24][25] Similar to our patient, all previously reported cases had a degree of dysphagia requiring endoscopic therapy. However, all cases had squamous papillomas without dysplasia, in contrast to our patient who was found to have ESCC.…”

A Case of Focal Dermal Hypoplasia (Goltze Syndrome) Masquerading as Lingual Tonsillar Hypertrophy

“…Oropharyngeal manifestations are less common, but can include papilloma on the tongue, tonsils, soft palate, hypopharynx, and larynx [3, 7]. Other case reports have also made note of squamos papilloma of the oesophagus [8]. There are 5 reports in the literature of oropharyngeal papillomatous lymphoid hyperplasia, and 4 of these occurred in young females as with our patient.…”

Rare and unusual case of familial focal dermal hypoplasia (Goltz syndrome) presenting to otolaryngology in the UK

“…This showed a marked reduction in papillomata with reported regurgitation without dysphagia after 4 months 10. Argon plasma coagulation technique for oesophageal papillomas in FDH has also been safely described in a child 11. Balloon-assisted radiofrequency ablation has also been described over a 10-cm area within the oesophagus with 4–5 mm projections achieving complete ablation on the second and third treatments 12…”