Sinonasal Hamartomas: From Nasal Chondromesenchymal Hamartoma to Respiratory Epithelial Adenomatoid Hamartoma. Report of six Cases and Review of the Literature

Sinonasal hamartomas are uncommon lesions of nasal and sinus cavities. Based on indigenous cellular components and characteristic histologic features, they are further classified into four entities: respiratory epithelial adenomatoid hamartoma (REAH), seromucinous hamartoma (SH), chondro-osseous and respiratory epithelial hamartoma (CORE), and nasal chondromesenchymal hamartoma (NCH). REAH, SH, and CORE are seen in adult patients, while NCH predominantly occurs in newborns and infants. Morphologically REAH and SH are composed of respiratory epithelium and seromucinous glands, CORE is related to REAH but with additional feature of chondroid and/or osseous tissue, and NCH is composed of chondroid and stromal elements but devoid of epithelial component. All four lesions can present as sinonasal mass lesions and with associated obstructive symptoms. Given the rarity of these lesions, diagnosis can be challenging, especially in unusual clinical scenario. In this study, we report six cases of sinonasal hamartoma, including one case of NCH, one case of CORE, two cases of SH, and two cases of REAH. All cases were from adult patients including four men and two women. We also review the literature of the clinical and pathologic features of these rare lesions.

Transformation from Sinonasal Seromucinous Hamartoma to Adenocarcinoma: A Case Report

<b><i>Objectives:</i></b> Sinonasal hamartomas are benign neoplasms composed of disorganized mature tissue elements. Epithelial variants include respiratory epithelial adenomatoid hamartoma (REAH) and seromucinous hamartoma (SMH). Malignant transformation of REAH is rarely reported; however, the malignant transformation of SMH to adenocarcinoma has not been described. We report the first case of a transformation from SMH to adenocarcinoma. <b><i>Methods:</i></b> The medical records of a patient presenting with sinonasal SMH with malignant transformation to adenocarcinoma were reviewed. The NCBI database was queried for the literature regarding SMH and malignant transformation of sinonasal hamartomas. <b><i>Results:</i></b> A 39-year-old man presented with a left nasal mass, nasal obstruction, and epistaxis. Computed tomography and magnetic resonance imaging demonstrated a nonaggressive and heterogeneous left nasal mass with involvement of the middle turbinate and posterior ethmoid sinuses. He underwent endoscopic sinus surgery with complete excision of the mass. Pathology revealed SMH with focal areas of transition to low-grade adenocarcinoma characterized by stromal invasion but no bony, perineural, or lymphovascular invasion. Adjuvant treatment was not recommended. Literature review revealed no reported cases of malignant transformation of SMH. <b><i>Conclusion:</i></b> We report the first case of malignant transformation of SMH. Patients with SMH must be counseled that there is an extremely rare and potentially unrecognized risk of malignancy that may influence treatment and postoperative monitoring.

CHONDRO-OSSEUS RESPIRATORY EPITHELIAL ADENOMATOID HAMARTOMA. CLINICAL CASE

Hamartomas of sinonasal tract are a very rare and poorly studied phenomenon, even within the framework of world medicine. In the classification of tumors of the head and neck according to the WHO, in the section of nasal papillomas, only respiratory epithelial adenomatoid hamartoma is mentioned, there is no information about the other types of nasal hamartomas. The peculiarity of this clinical case is the difficulty in verifying the type of hamartoma of the sinonasal tract (respiratory epithelial adenomatoid, seromucinous, chondromesenchymal or chondro-osseous respiratory epithelial adenomatoid), respectively, and in determining the tactics of subsequent treatment, due to the rarity of this type in the world and the possibility of malignant transformation. A 15-year-old patient was diagnosed with chondro-osseous respiratory epithelial adenomatoid hamartoma. According to the literature, only 12 such clinical cases were recorded in the world. The absence of specific radiological and laboratory markers, the clinical picture of the disease similar to other diseases of the nose and paranasal sinuses - all this complicates early diagnosis, and emphasizes the need to include this type of neoplasm in the differential diagnostic range in children and adults with masses of the nasal cavity.

Non-resolving acute sinusitis due to nasal hamartoma: a case report

The paranasal sinuses are closely related to vital structures, and therefore, rhinosinusitis may lead to various dangerous complications. Nasal chondromesenchymal hamartoma is a rare benign lesion of the sinonasal tract in children and adolescents. We report the case of a 15-year-old patient with antibiotic-refractory acute sinusitis in whom a nasal tumor was identified and histopathologically diagnosed as a hamartoma. Therefore, the tumor was removed surgically to alleviate the patient’s symptoms. This case highlights the necessity of investigating the underlying cause if conventional treatment fails to resolve a mild disease like acute sinusitis. To the best of our knowledge, this is the first reported case of a chondro-osseous respiratory epithelial adenomatoid hamartoma presenting with acute sinusitis.

Contents Vol. 33 no. 2 July – December 2018

EDITORIAL 4 A Dozen Years, A Dozen Roses Lapeña JF ORIGINAL ARTICLES 6 Efficacy of Clarithromycin Versus Methylprednisolone in the Treatment of Non-Eosinophilic and Eosinophilic Nasal Polyposis: A Randomized Controlled Trial Gammad JC, Chua AH, Templonuevo-Flores CS 14 Simulation Platform for Myringotomy with Ventilation Tube Insertion in Adult Ears Chan AL, Carrillo RJD, Ong KC 21 Prevalence of Supraorbital Ethmoid Air Cells among Filipinos Carlos ALC, Gelera JE 24 Recurrent Laryngeal Nerve Paralysis and Hypocalcemia in Superior to Inferior Compared to Inferior to Superior Dissection Approaches in Thyroidectomy Yu RD 28 Pathologic Laryngoscopic Findings, Number of Years in Teaching, and Related Factors among Secondary Public-School Teachers in Bacolod City, Negros Occidental Mundo NP, Vinco VV 32 Tracheal Diameter Estimates Using Age-Related Formula Versus Radiographic Findings: Which Approximates the Actual Tracheostomy Tube in Pediatric Patients? De Guzman ICS CASE REPORTS 37 Lethal Midline Granuloma in a 15-Year-Old Girl: A Diagnostic Dilemma Gonzales ICA 41 Tuberculosis of the Temporomandibular Region Santos JM, Reala ET 45 Crab Shell Impaction in the Larynx with Aphonia Parekh NM, Kashyap PR SURGICAL INNOVATION AND INSTRUMENTATION 48 PHONETOVOX: A Novel Device for Alaryngeal Speech Econ ME, Soriano RG 53 Can Modified Laryngosternopexy (Laryngoclaviculopexy) Project the Larynx Anteriorly? Carrillo RJC, Lapeña JF FEATURED GRAND ROUNDS 56 Cleft Beyond the Lip and Palate: A Bilateral Tessier Cleft Canta LAB FROM THE VIEWBOX 60 Pulsatile Tinnitus Due to a Sigmoid Sinus Diverticulum and/or Dehiscence Yang NW UNDER THE MICROSCOPE 62 Respiratory Epithelial Adenomatoid Hamartoma Carnate JM, Abelardo AD PASSAGES 64 Armando T. Chiong, Sr. (1930 - 2018) Jamir JC 65 Carlos P. Dumlao (1950 - 2018) Pontejos AQY