BACKGROUND:

Acute oral angioedema is well documented in young pediatric liver-transplanted patients. This is a report of an identified novel entity of long-standing oral mucosal lesions (OML) in children who had undergone solid organ transplantation.

METHODS:

In conjunction with routine medical follow-ups, all solid organ-transplanted children were examined annually for OML.

RESULTS:

Eight children developed a novel entity of long-standing OML after solid organ transplantation. Seven of the eight children had undergone liver transplantation whereas one child was kidney transplanted. The children received tacrolimus immunosuppression after transplantation. All recipients presented with multiple spherical nodules on the dorsum of the tongue, which later on displayed a fissured appearance. Most patients also presented with mucosal tags or ridges and swollen lips similar to those found in patients with orofacial granulomatosis. In addition, most patients had a clinical history of immediate-onset food-induced allergic reactions, including transient angioedema, after transplantation, but this clinical manifestation appeared separately from the long-standing lesions.

CONCLUSION:

A novel entity of long-standing OML that share some features with orofacial granulomatosis has been identified in children after solid organ transplantation.