Rare Lesion Research Articles

A large gastric inflammatory fibroid polyprevealed by an upper gastrointestinal bleeding: a case report.

Inflammatory fibroid polyps (IFPs) are rare submucosal lesions that usually arise in the stomach. It represents 0.1% of all gastric polypoid lesions and are frequently asymptomatic. It's usually revealed by an abdominal pain. Gastrointestinal bleeding can occur when the polyp is ulcerated. We report a case of a 72-year-old female with an IFP of the prepyloric region. The head of the polyp was thirty-five millimeters in diameter. It was revealed by an upper gastrointestinal bleeding and epigastric pain. An endoscopic resection was performed.

Giant benign mesenteric cysts (mesothelioma and lymphangioma): A report of two cases

Introduction and importanceMesenteric cysts are a rare group of intra-abdominal tumors located in the mesentery or omentum, most of this lesion are asymptomatically but occasionally presents with non-specific symptoms, which makes diagnosis difficult. Case presentationWe present two cases of giant mesenteric cyst in our Institution, the first case is a women of 23 years with previous gynecologic surgery with chronic abdominal pain and large abdominal mass CT showed a giant intraabdominal 30 × 25 × 15 cm, the patient went to laparotomy and a large mesenteric tumor found. The Histopathology reported a Benign cyst mesothelioma (BCM). The second case is a 47 year male with intermittent abdominal pain and abdominal CT shows a 20x21x15 cm tumor. Laparotomy was performed. Histopathology reported a large lymphangioma. Clinical discussionBCM tumors are rare intrabdominal lesions often diagnosed in premenopausal women. Its etiopathogenesis is still unclear, associated risk factors are endometriosis, pelvic inflammatory disease and previous pelvic surgery. Lymphangiomas are an infrequent cyst tumors. It results from a failure in the evolutionary development of the lymphatic system, often localized in the head and neck and its localization in the abdomen is even rare. The definitive diagnosis is made by histopathology. ConclusionMesenteric cysts diagnosis if often challenge due to nonspecific symptoms and significant overlap in imaging appearance between the different entities, its diagnosis should be in mind when encounter a cystic abdominal tumor, surgical complete removal of this tumors is the optimal treatment to prevent recurrence and complications.

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

BackgroundWe 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 presentationWe 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.ConclusionSeromucinous 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.

Calcifying Odontogenic Cyst: Report of Two Cases Report in a Different Stage of Evolution

Odontogenic tumors with calcified components contribute substantially to the diversity in the field of oral surgery. Among these, the Calcifying Odontogenic Cyst (COC), also known as Gorlin's cyst, stands out. It is a rare odontogenic lesion, first described by Gorlin in 1962. It has a vast variety of radiological, clinical, biological behaviors and histopathological features. In this article, two cases of Calcifying Odontogenic Cyst (COC) treated successfully with surgical interventions at oral Medicine and Oral Surgery department at the academic dental Clinic of Monastir, in order to underline clinical, radiological and histological features of this cyst and highlights the interest of detailed complementary investigations particularly anatomopathological ones to precisely define the diagnosis.

Low-Grade Appendiceal Mucinous Neoplasm vs. Appendiceal Diverticulum: Distinction with Histomorphologic Features

Background: Low-grade appendiceal mucinous neoplasms (LAMNs) are rare lesions of the vermiform appendix and characterized by mucinous epithelial proliferation, extracellular mucin, and the absence of destructive invasion. Appendiceal diverticulum (AD) is also an uncommon condition that may be challenging to differentiate from acute appendicitis when it is superimposed by diverticulitis or perforation. Some recently published studies emphasized that complicated AD with mucosal hyperplasia can be confused with LAMNs, leading to overdiagnosis. The present study aimed to determine the histopathological features which can be used in the differential diagnosis of LAMNs and ADs, particularly complicated diverticula, in a large cohort. Methods: Cases comprising LAMNs and ADs diagnosed between 2011 and 2021 were included in the study. All cases were evaluated for the epithelial lining, the wall of the lesions, and the presence of cellular or acellular mucin, with its localization in terms of level and site of involvement within the appendix also recorded. Results: The hypermucinous epithelium characteristic of LAMNs, fibrosis, and calcification in the wall and the absence of lamina propria and muscularis mucosa proved to be the most discriminatory features in the differential diagnosis of LAMNs and ADs. Conclusions: The distinction between mucinous neoplasia and its mimics is critically important, since mucinous neoplasia requires surveillance imaging and potential surgery or chemotherapy depending on the extent of the disease, whereas non-neoplastic lesions are treated by an appendectomy and require no future intervention or surveillance.

The Occurrence and Outcomes of Cemento-Osseous Dysplasias (COD) in the Jaw Bones of the Population of Lower Silesia, Poland.

Background: Cemento-osseous dysplasias (CODs) are rare lesions of the jawbone. Their occurrence, localization, type, size, and shape can vary between cases. This fibro-osseous lesion is typically found in the jaw near tooth-bearing areas and is often asymptomatic, discovered incidentally, and may be associated with the periapical region of the teeth. In rare cases, COD can lead to secondary bone osteomyelitis. Currently, there is limited information in the literature on the occurrence and characteristics of COD. This paper's main aim was to focus on the authors' COD experience in the lower Silesian area. Methods: A retrospective evaluation of radiographies (RTG-Panx, cone-beam computed tomography (CBCT)) was conducted on patients treated, diagnosed, or consulted by the authors. A statistical correlation analysis was made to establish any relationship within the gathered data. Results: COD is predominantly an incidental finding in the mandibular bone near tooth apices. It is most commonly diagnosed in females. Both CBCT and panoramic radiographies are generally sufficient for diagnosing the lesion. COD rarely requires treatment. Conclusions: COD lesions are mostly discovered incidentally during routine radiographies or cone-beam computed tomography (CBCT) scans. In most cases, clinical and radiological monitoring is sufficient, along with evaluating the teeth's response to cold stimuli and assessing the surrounding bone structures. Biopsies or tooth extractions are seldom necessary. When oral hygiene is well-maintained and no periapical inflammation is present, COD lesions typically remain asymptomatic.

A case of acute idiopathic maculopathy in both eyes with peripheral vascular occlusion

BackgroundThis study aimed to present a case of bilateral symmetric onset of acute idiopathic maculopathy after high fever with peripheral vascular occlusion.Case presentationA 54-year-old yellow female experienced acute binocular visual acuity decrease after 1 day of high fever, and binocular visual acuity decreased to index or anterior. OCT in the external hospital showed dome-shaped cortical detachment in the symmetrical macular area of both eyes with subretinal fluid, discontinuity in the ellipsoid zone and the chimera. The patient autonomously administered antibiotics and non-steroidal anti-inflammatory agents, including amoxicillin and ibuprofen. Following a three-day period, the patient’s visual acuity demonstrated significant improvement. Additionally, macular edema demonstrated a notable reduction as indicated by optical coherence tomography, while the presence of peripheral retinal vascular occlusions was also observed on fluorescein fundus angiography. At the onset of 6 days, oral hormone therapy was given in the outside hospital, with no significant improvement in visual acuity.23 days after the onset of the disease, the patient was admitted to our hospital, and was finally diagnosed as acute idiopathic maculopathy combined with the imaging findings of FFA, ICGA and OCT. During the follow-up, the visual acuity of both eyes improved spontaneously.ConclusionsThis case is a rare acute idiopathic macular lesion with bilateral involvement, accompanied by highly symmetrical peripheral retinal vascular occlusion in both eyes, which deepens our understanding of acute idiopathic maculopathy with a view to providing guidance for subsequent clinical practice.

Solitary Peutz-Jeghers polyp harboring a focus of high-grade dysplasia in the colon: a case report and literature review.

A solitary Peutz-Jeghers (PJ) polyp is a rare hamartomatous lesion without an associated PJ syndrome. However, little is known regarding malignancy arising in solitary PJ polyps. Here, we report a case of a solitary colonic PJ polyp with focal dysplasia. A 45-year-old asymptomatic man underwent total colonoscopy following a positive fecal occult blood test. The patient had no history of mucocutaneous pigmentation or family history of PJ syndrome. A 20mm erythematous pedunculated polyp was observed in the sigmoid colon. Magnified endoscopy revealed a tubular or branch-like pit pattern with localized areas of irregular pits. These findings were suggestive of colorectal adenoma with high-grade dysplasia, and endoscopic mucosal resection was performed. Histopathological examination revealed arborizing proliferation of hyperplastic epithelia with smooth muscle bundles. In addition, a small number of irregular crypts with high-grade dysplasia were observed in the hyperplastic epithelium. Based on these histological findings, we finally diagnosed the patient with a solitary colonic PJ polyp with high-grade dysplasia. The present case suggests that solitary colonic PJ polyps may harbor dysplastic changes and require pathological evaluation with en bloc resection of the polyps.

Clinical Characteristics and Genomic Profile of Malignant Proliferating Trichilemmal Tumor: A Systematic Review of the Literature.

Malignant proliferating trichilemmal tumors (MPTT) are rare skin lesions arising from the outer sheath of the hair follicle root. Because of their rarity and difficulty in pathologic identification, these skin lesions are often initially misdiagnosed. After a literature review, we found initial clinical or histopathological misdiagnosis has an associated 10.4-fold increase in recurrence and 2.18-fold increase in deaths.

A Rare Neck Lesion in Children: Bilateral Chondrocutaneous Branchial Remnant.

Cervical chondrocutaneous branchial remnant is a rare congenital developmental anomaly typically located on the lateral neck. Histologically, it has the appearance of an accessory tragus demonstrating a central cartilaginous core with surrounding fibrosis located in the subcutaneous tissue. In order to address the clinical and therapeutic aspects of cervical chondrocutaneous branchial remnants in children, a 6-year-old boy who presents with bilateral hard cervical masses at the lower region of the neck is presented. Chondrocutaneous branchial remnant is a very rare cervical lesion in children, and surgical excision is the only choice of treatment. Cervical chondrocutaneous branchial remnant has been associated with a variety of congenital anomalies, particularly involving the auditory, cardiovascular, and visual systems. Despite bilateral occurrence of cervical chondrocutaneous branchial remnant, our patient had no cardiac or genitourinary anomalies.

Central giant cell granuloma of mandible: a case report

Central Giant Cell Granuloma(CGCG) and giant cell tumour of jawbone are grouped underneath same umbrella but with distinct clinical behavior. CGCG is a rare benign lesion of the jaws distinguish by the presence of multinucleated giant cells within a fibrous stroma. The pathogenicity of CGCG still remains an enigma, which demands the differentiation from other look-alike lesions in order to have proper diagnosis and treatment planning. In this article, a case of CGCG in a 30-year-old female patient who presented with complaints of gradually increasing swelling and discomfort in the mandible is described.

Nasal dermoid sinus cyst with intracranial extension and atretic sinus tract in an adult patient

Nasal dermoid sinus cyst (NDSC) is a rare congenital lesion due to cephalic neural tube defect. It may also have been seen in other parts of the body. NDSC constitutes 1–3% of all dermoid cysts and 11–12% of those in the head and neck. A NDSC is located in the median line of the nasofrontal region from the glabella to the columella. The differential diagnosis based on location includes epidermal inclusion cyst, glioma, meningomyelocele, meningoencephalocele, teratoma, teratoid cyst, thyroglossal duct cyst, branchial cleft cyst, and benign lymphadenopathy. Intracranial extension causes serious complications such as meningitis, osteomyelitis, abscess formation, hydrocephalus, seizures, and personality changes. All patients with NDSC should be considered to have a potential intracranial extension and should, therefore, undergo pre-operative radiological evaluation. Herein, the authors report an unusual adult patient with NDSC with intracranial extension.

A retrospective analysis of alimentary tract duplications in pediatric patients: a 14-year single-center experience.

Alimentary tract duplications (ATDs) are rare congenital lesions often associated with anomalies such as spinal, urinary and GI tract malformations. The purpose of this study was to report the experience of a single center with ATDs in children, focusing on the natural history, associated malformations, and their impact on patient management. We performed a retrospective analysis over 14years, collecting prenatal, clinical, surgical, and follow-up data. We focus on associated anomalies prenatal and postnatal management, and outcomes. Sixty-three patients with ATD (thirty-six females, twenty-seven males, aged 1 day to 14years) were enrolled in this study. Prenatal diagnosis was made in 22 patients (35%), of whom 8 showed compression signs. Elective surgery was performed at a mean age of 1.5years in prenatally diagnosed cases. The others presented symptoms at a mean age of 5.2years (55.5%) or were detected incidentally (9.5%) at a mean age of 10.7years. In four patients (6.3%), we identified multiple duplications, and ten cases (15.8%) were found with associated anomalies. The wide spectrum of clinical appearance of ATDs and a comprehensive knowledge of human embryology might define surgical management, which should always be patient-tailored and respectful of the child's development.

Epidermal Inclusion Cyst Presenting as Prepatellar Bursitis

An epidermal inclusion cyst is one of the rare benign lesions that can occur around the knee and can mimic prepatellar bursitis. They can occur anywhere in the body, but more prevalent sites are the face, neck, trunk, behind the ears, and palmoplantar region. In the present case, a 52-year-old man with epidermal inclusion cyst presented to the surgery outpatient department (OPD) with symptoms clinically mimicking as prepatellar bursitis, occurring at a rather unusual site, ie; the anterior aspect of the left knee. The possibility of an epidermal inclusion cyst should always be considered in an atypical soft tissue mass when presenting around the knee. Histopathological examination confirms the diagnosis.

Isolated Dorsal Distal Radio-Ulnar Dislocation: About A Case and Review of the Literature

Isolated acute dislocation of the distal radioulnar joint is a rare lesion, and in 50 % of cases goes unrecognized; it may be palmar or dorsal. Its diagnosis is suspected when the wrist is traumatic, painful, with limited pronosupination and no fracture on radiological examination. Treatment is aimed at preventing the development of chronic instability and/or arthrosis, and consists of reduction followed by immobilization.

Superficial Papular Neuroma: Two Cases of a Distinctive Dermal Neoplasm.

Superficial papular neuroma is a rare cutaneous spindle cell lesion, with only 5 cases reported in 2 studies. We document 2 additional cases in a 45-year-old man and a 43-year-old woman (the first case identified in a woman). Clinically, superficial papular neuromas appear as a single, non-specific papule on the head, neck, or back. Histologically, these lesions are within the papillary and superficial reticular dermis, with nerve-like structures composed of bland spindle-shaped cells intersecting normal tissue with mild reactive acanthosis. The cells are SOX10 positive with neurofilament protein staining multiple axons within. The nerve-like structures in this study were occasionally surrounded by a thin rim of CD34 positivity, with no epithelial membrane antigen staining, similar to normal sensory neurons. Superficial papular neuroma are rare benign neoplasms with no reports of recurrence, even when incompletely excised.

Myoid Hamartoma of the Breast With HMGA2 Rearrangement and Associated In-Situ and Invasive Carcinoma: Case Report and Review of Literature.

Myoid hamartoma of the breast is an uncommon benign breast neoplasm. We describe an unusual example of an in-situ and invasive carcinoma arising in a myoid hamartoma. We also describe the unique molecular findings in the myoid hamartoma and review the pertinent literature. In radiology studies, the mammogram showed increased density/attenuation, and the ultrasound revealed a change in the overall echogenicity with hyperechoic and anechoic areas. The stromal compartment contained smooth muscle bundles and adipose tissue on microscopic examination. The stromal cells showed HMGA2 gene rearrangement by fluorescence in-situ hybridization analysis and overexpression of HMGA2 by immunohistochemistry. The breast myoid hamartoma is likely underpinned by HMGA2 gene rearrangement and HMGA2 protein overexpression, which can be used as an ancillary test to diagnose this rare breast lesion. Although most myoid hamartomas have a benign clinical course, the likelihood of malignant transformation should always be considered.

Pulmonary aortic endocarditis in patient with patent ductus arteriosus

Introduction: Right-sided heart endocarditis is uncommon, comprising only 5-10% of all infective endocarditis cases. The infective process may be isolated to pulmonary valve or concomitantly involve other valves. Prompt diagnosis by transthoracic echocardiography, aggressive antibiotics from the beginning, and surgical removal of the vegetation for infective endocarditis help prevent the risk of multiorgan failure and fatal pulmonary embolism. Case summary: A 38-year-old female, the medical history was normal, gave birth naturally at a district hospital 3 months ago. She admitted to the hospital because of fever and continuous shortness of breath 1 month before admission. Echocardiography detected: vegetations (10x23mm) on left pulmonary aortic and patent ductus arteriosus. A CT scan showed a mixed-echo mass in the left pulmonary artery. The patient had surgery to remove the vegetation and close the patent ductus arteriosus. The patient was stable after surgery and discharged 2 weeks after surgery. Conclusion: Pulmonary aortic vegetations and patent ductus arteriosus are rare lesions and high risk of death. The diagnosis should be considered in any febrile, septic patient with congenital heart disease. Surgery removal of vegetations and aggressive antibiotic treatment should be performed together to improve the outcome.

Solid pancreatic masses: What’s hidden beneath? Insights into rare pancreatic lesions

The diagnostic approach to solid pancreatic masses has significantly evolved from the era when a focal pancreatic mass was almost synonymous to pancreatic ductal adenocarcinoma, to a wide spectrum of pancreatic lesions, some of which have good prognosis. With the advent of advanced diagnostic tools, particularly refined imaging and tissue acquisition techniques, a broader spectrum of differential diagnoses has been recognized, encompassing conditions ranging from neuroendocrine tumors or inflammatory masses, to rare entities like metastatic clear cell sarcoma or solitary fibrous tumors. We herein discuss case reports of some rare pancreatic lesions, which were diagnosed by combining clinical and imaging features and endoscopic ultrasound-guided tissue sampling and confirmed on surgical specimens. Further reports on these rare pancreatic tumors will contribute to a better understanding of their pathogenesis and effective management.

Primordial Odontogenic Tumor: A Decade Post-Description Systematic Review.

Primordial odontogenic tumor (POT) is a rare benign tumor arising from odontogenic epithelium and ectomesenchyme. It typically presents in children and young adults. POT is often found in the posterior mandible and frequently presents as asymptomatic swelling. A systematic review of the literature was conducted to comprehensively analyze the clinicopathologic features of this rare entity over the past ten years. A systematic review of POT case series and case reports following the Preferred Reporting Items for Systematic Reviews and Meta-analyses (PRISMA) statement guidelines was performed. Data on demographics and clinical characteristics, including age, sex, clinical presentation, duration of the lesion, location, and radiographic and histological features, were extracted. A quantitative description of immunohistochemical studies reported in the literature was also performed. Treatment, a follow-up period, and recurrence were collected for analysis. This review included 26 studies involving 36 POT cases. Patients often presented with asymptomatic swelling at a median age of 12 years, with a male-to-female ratio of 1.18:1. The posterior mandible was the most commonly affected site, while three cases were noted in the anterior region exclusively in the maxilla. Most lesions appeared as unilocular radiolucencies with well-defined borders; however, five cases exhibited fine trabeculation or radiopacities. The primary histological features observed in POT included ectomesenchymal stroma lined by columnar cells with nuclear reverse polarity. Most cases were treated through enucleation and curettage (50.0%), followed by tumor excision (36.1%). Only one case demonstrated recurrence among the 29 cases with known follow-up information. This study offers comprehensive and current descriptive data on POT, enhancing the ability of clinicians and pathologists to accurately identify these rare lesions and thereby avoid misdiagnosis and inappropriate management.