Seromucinous hamartoma with unique clinical and histopathological features: a case report and review of the literature.

IF 0.9 Q3 MEDICINE, GENERAL & INTERNAL Journal of Medical Case Reports Pub Date : 2024-11-24 DOI:10.1186/s13256-024-04843-2
Mikhael Makhoul, Nadim Khoueir, Myriam Khneisser, Hussein Nassereddine
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Abstract

Background: We present this case to highlight the importance of considering seromucinous hamartoma in the differential diagnosis of nasal cavity lesions, particularly due to its rarity and potential for being mistaken for more aggressive pathologies. Seromucinous hamartoma, although benign, can exhibit clinical and histological features that overlap with those of malignant tumors, posing a diagnostic challenge. This case is especially noteworthy due to the unusual presentation of sebaceous differentiation within seromucinous hamartoma, a feature not previously documented in the literature. Recognizing such rare entities is crucial for ensuring appropriate patient management and avoiding unnecessary interventions.

Case presentation: We report a unique case of a 41-year-old Caucasian man with seromucinous hamartoma, presenting with chronic left nasal obstruction and recurrent mild epistaxis. Nasal endoscopy revealed a well-defined, multilobulated lesion in the left nasal cavity. Computed tomography scans confirmed an anteromedial polypoid lesion, 31 mm × 15 mm. The lesion was endoscopically resected without complications, with no recurrence at 6-month follow-up. Pathological examination showed a filiform polypoid lesion with clusters of seromucinous glands, ducts, and tubules, and no invasive growth patterns. Immunohistochemical studies revealed distinct epithelial membrane antigen (EMA) and p63 staining patterns. Notably, mature sebaceous gland formation interspersed with seromucinous glands was observed, a novel finding in seromucinous hamartoma. Recognizing seromucinous hamartoma is crucial to avoid unnecessary treatments, and it should be included in differential diagnoses of nasal cavity lesions.

Conclusion: Seromucinous hamartoma is a rare and benign nasal cavity lesion that presents significant diagnostic challenges due to its potential to mimic more aggressive pathologies. This case highlights the importance of including seromucinous hamartoma in the differential diagnosis of sinonasal lesions, particularly when unusual histological features, such as sebaceous differentiation, are present.

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具有独特临床和组织病理学特征的血清粘液性火腿肠瘤:病例报告和文献综述。
背景:我们介绍这个病例是为了强调在鼻腔病变的鉴别诊断中考虑血清粘液瘤的重要性,特别是由于其罕见性和被误诊为更具侵袭性病变的可能性。血清粘液瘤虽然是良性的,但其临床和组织学特征可能与恶性肿瘤重叠,这给诊断带来了挑战。本病例尤其值得注意的是,血清粘液性仓壁瘤内皮脂腺分化的表现不寻常,而这一特征在以前的文献中从未有过记载。识别这种罕见的实体对于确保适当的患者管理和避免不必要的干预至关重要:我们报告了一例独特的病例,患者是一名 41 岁的白种男子,患有血清粘液性仓壁瘤,表现为慢性左鼻塞和反复轻度鼻衄。鼻内窥镜检查发现左鼻腔内有一个界限清楚的多叶状病变。计算机断层扫描证实为前内侧息肉样病变,大小为 31 毫米×15 毫米。病灶经内窥镜切除,无并发症,随访6个月无复发。病理检查显示,病灶为丝状息肉样病变,内有成群的血清粘液腺、导管和小管,无浸润性生长。免疫组化研究显示,上皮膜抗原(EMA)和 p63 染色模式各不相同。值得注意的是,成熟的皮脂腺形成与浆液粘液腺穿插在一起,这是浆液粘液性火腿肠瘤的一个新发现。识别血清粘液性火腿肠瘤对于避免不必要的治疗至关重要,应将其纳入鼻腔病变的鉴别诊断中:结论:血清粘液瘤是一种罕见的鼻腔良性病变,由于其有可能模仿更具侵袭性的病变,因此给诊断带来了巨大挑战。本病例强调了将血清粘液瘤纳入鼻窦病变鉴别诊断的重要性,尤其是当出现皮脂腺分化等异常组织学特征时。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
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来源期刊
Journal of Medical Case Reports
Journal of Medical Case Reports Medicine-Medicine (all)
CiteScore
1.50
自引率
0.00%
发文量
436
期刊介绍: JMCR is an open access, peer-reviewed online journal that will consider any original case report that expands the field of general medical knowledge. Reports should show one of the following: 1. Unreported or unusual side effects or adverse interactions involving medications 2. Unexpected or unusual presentations of a disease 3. New associations or variations in disease processes 4. Presentations, diagnoses and/or management of new and emerging diseases 5. An unexpected association between diseases or symptoms 6. An unexpected event in the course of observing or treating a patient 7. Findings that shed new light on the possible pathogenesis of a disease or an adverse effect
期刊最新文献
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