Outcomes and imaging findings of respiratory epithelial adenomatoid hamartoma: a systematic review

Abstract

BackgroundRespiratory epithelial adenomatoid hamartoma (REAH) is a recently classified histopathologic diagnosis often identified incidentally following endoscopic sinus surgery (ESS) for presumed chronic rhinosinusitis. Limited data exist defining preoperative imaging features and surgical outcomes. The purpose of this study is to examine characteristic imaging findings of REAH and postoperative olfactory and recurrence outcomes.MethodsA systematic review was performed according to the Preferred Reporting Items for Systematic Reviews and Meta‐Analysis (PRISMA) guidelines on articles published from 1995 to present. PubMed, EMBASE, and Ovid MEDLINE databases were queried for studies pertinent to imaging findings of REAH and surgical outcomes. Quality of articles was assessed using the Methodological Index for Non‐Randomized Studies (MINORS).ResultsA total of 294 articles were identified, with 15 meeting inclusion criteria. Seven articles assessed both imaging findings and surgical outcomes. Three articles focused exclusively on imaging, whereas 5 examined surgical outcomes. Olfactory cleft (OC) widening greater than 10 mm on computed tomography (CT) was characteristic of REAH. A total of 441 patients with REAH were included; 221 patients (50.1%) had concurrent nasal polyposis, whereas 154 patients (34.9%) had isolated REAH. Surgical intervention ranged from simple excision to complete ESS. Sixty‐five percent (65%) of patients reported improved olfaction; 4.1% of patients recurred with follow‐up ranging from 4 months to 5 years.ConclusionA widened OC may suggest the presence of REAH. This disease process has been identified in patients with nasal polyposis or encountered as an isolated lesion. Targeted surgery may result in improved olfaction and a low likelihood of recurrence, though long‐term prospective studies are necessary.

Is Respiratory Epithelial Adenomatoid Hamartoma Related to Central Compartment Atopic Disease?

“…REAH frequently occurs in the central compartment of the nasal cavity, with the posterior nasal septum and OC being the most commonly reported locations. 9,13 Although location reported varies by study and sampling method, many authors have identified an association with middle turbinate lateralization picked up as an enlarged, opacified OC on computed tomography (CT) imaging. Seven out of 10 studies assessed for imaging findings in a recent systematic review found evidence of OC enlargement/involvement, with one study reporting that OC width greater than 10 mm resulted in 88% sensitivity for REAH.…”

“…This theory is further supported by the finding that the main treatment for CCAD-conservative resection and sculpting of abnormal appearing mucosa down to as normal appearing anatomy as possible without exposing underlying bone-has resulted in low rates of recurrence for both REAH and CCAD. [5][6][7]9,13,19 Further studies will be necessary to investigate the pathogenesis of REAH and related disease in the central compartment of the nasal cavity. This study was limited by its retrospective nature, as the true rate of allergy could not be fully assessed in some patients due to lack of documentation of allergy status.…”

“…Treatment with conservative surgical resection has resulted in low rates of recurrence, though there are no evidence-based treatment recommendations. 9 Therefore, establishing the diagnosis of REAH is of critical importance in order to avoid unnecessarily radical surgery.…”

“…Evidence that REAHs express higher levels of fractional allelic loss compared to polypoid sinus tissue, as well as reported cases of malignant tumors occurring in association with REAH, suggest that REAH may be a neoplastic process. [9][10][11] However, the association of REAH with chronic sinonasal inflammatory disorders, prior history of sinus surgery, and asthma supports the theory that REAH is a reactive (hyperplastic) process occurring in the setting of chronically inflamed or damaged mucosa. 5,7,12 Furthermore, the physiologic basis for the frequent occurrence of REAH in the superior nasal septum and olfactory cleft (OC) is not well understood.…”

“…5,7,12 Furthermore, the physiologic basis for the frequent occurrence of REAH in the superior nasal septum and olfactory cleft (OC) is not well understood. 1,2,9,13 Recent evidence has established isolated polyposis in this central compartment of the nasal cavity to be a distinct form of nasal inflammatory disease associated with a high incidence of allergic sensitization. [14][15][16][17][18][19] The purpose of this study was to report our series of pathologically diagnosed REAH and to explore other factors that may explain the etiology of REAH.…”