Oral White Sponge Nevus in a Four-Generation Family: Case Report

Abstract

Bijeli spužvasti nevus ili madež (WSN) rijedak je dobroćudni autosomno dominantni poremećaj obično poput mekane, bijele, spužvaste naslage na mukoznoj membrani, najčešće oralnoj. Pojavljuje se odmah nakon rođenja ili u djetinjstvu, bez obzira na spol. U prikazu je opisan slučaj dječaka u dobi od četiri godine i njegova 42-godišnjeg oca. Obojica su se žalili na bezbolne bijele lezije u usnoj šupljini. Obiteljska i porodična anamneza otkrila je da od osamnaestero članova njihove porodice njih jedanaestero (61,11%) ima WSN. Kliničkim pregledom otkrivena je difuzna, preklopljena, spužvasta bijela naslaga koja je obostrano zahvatila bukalnu sluznicu i druge dijelove oralne sluznice. Histološkim pregledom ustanovljena je akantoza, hiperkeratoza spongioza i eozinofilna kondenzacija u perinuklearnom području spinoznog sloja. Dijagnoza WSN-a postavljena je na temelju pozitivne porodične anamneze tijekom četiriju generacija, te kliničkih i histoloških značajki. Za WSN nema specifične terapije. Iako je bijeli spužvasti madež rijedak, potrebno ga je uključiti u diferencijalnu dijagnozu bijelih lezija u djetinjstvu, posebice u slučaju pozitivne obiteljske anamneze.The aim of the present article was to discuss two different case reports of the extra-oral odontogenic cutaneous sinus tracts with emphasis on their diagnoses and treatment plans. Odontogenic cutaneous sinus is a rare but well documented condition. It is usually misdiagnosed as a local skin lesion and mistreated by multiple surgical excisions and/or systemic antibiotics with eventual recurrence. This is because the primary dental etiology is overlooked. We have described two cases of extraoral cutaneous sinus tract of an odontogenic origin. In both cases there were complaints of pain and purulent/hemorrhagic discharge from the lesions. Clinical and radiological examination showed inactive teeth, which were infected periapically in both cases. Non-surgical endodontic treatment was successful in Case 1. Surgical intervention was performed in Case 2. Both patients responded well and healing was uneventful. An understanding of the pathogenesis of cutaneous sinus tract arising from dental infection will lead to proper early diagnosis and successful treatment