Abstract
Respiratory epithelial adenomatoid hamartoma (REAH) is a benign tumor which most frequently appears as a unilateral or bilateral polypoid mass in the nasal cavity. As REAH often presents with symptoms similar to those of chronic rhinosinusitis (CRS), it is often erroneously diagnosed and treated as CRS.
Herein, we report a case of REAH, which we miss-diagnosed preoperatively as a severe case of ECRS according to the criteria of the JESREC study, however, the postoperative histopathological examination revealed the diagnosis of REAH.
A 26-year-old woman with a 4-year history of bronchial asthma presented to us with a one and a half-year history of hyposmia and hyper-rhinorrhea. At the first consultation at our clinic, examination revealed bilateral nasal polyps. CT examination revealed a diffuse shadow of soft tissue density in the olfactory cleft and ethmoid sinuses. The peripheral blood eosinophilic count was elevated to 7.3%. Consequently, we made the preoperative diagnosis of severe ECRS according to the criteria of the JESRAC study, and performed endoscopic pansinusectomy. The surgical specimen, which consisted of the hypertrophic mucosal membrane including the cystic lesion in the olfactory cleft and polyps in the middle meatus, was examined histopathologically. Histopathology revealed poor infiltration by eosinophils and proliferation of a large number of glands within the mucous membrane. In addition, the glands were lined by simple layers and the lumen were filled with mucinous or eosinophilic amorphous material. Based on the findings, we made the conclusive diagnosis of REAH. The postoperative progress has been satisfactory, with the patient showing no evidence of recurrence of the disease until date.
We had difficulty in diagnosing ECRS preoperatively based on the present diagnostic criteria of ECRS, because preoperative histopathology is not an essential diagnostic criterion now. In the case of REAH, complete extirpation of lesions may necessitate no additional treatments, because of the low frequency of recurrence of the disease. We must keep in mind the possibility of the diagnosis of REAH when dealing with patients with CRS. It is important to obtain the information about the histopathology of the disease before and/or after operation.
