Hybrid Lesion Research Articles

Congenital Pulmonary Hybrid Lesions: Clinical Presentation, Surgical Management, and Outcomes.

Congenital pulmonary airway malformation (CPAM) lesion with an aberrant systemic blood supply is considered a "hybrid" lesion (HL). The aim of the study was to report authors' experience in the presentation, surgical management, and outcomes of HL. A retrospective cohort study of HL patients followed from 2000 to 2022 in a single institution was done. Clinical and radiographic data were collected from the medical records. Over the study period, 39 patients with HL were identified. HL with CPAM type 2 was more common than type 1 (74% vs. 26%), and intralobar sequestrations were more prevalent than extralobar sequestrations (ELS) (79.5% vs. 20.5%). HL with CPAM type 2 had higher rates of co-morbidities and higher likelihood to present with respiratory symptoms after birth. HL with ELS had smaller feeding vessels (all < 4mm) which resulted in lower rates of open thoracotomy approach for resection. The variability in HL patient presentation and outcomes is correlated with the size and the hybrid composition.

The ten-year evaluation of clinical characteristics in congenital lung anomaly in pediatrics; a retrospective study in North of Iran

IntroductionCongenital lung anomalies (CLA) are a group of anomalies, including congenital cystic adenomatoid malformation (CCAM), bronchopulmonary sequestrations (BPS), congenital lobar emphysema (CLE), and bronchogenic cysts (BC). The prevalence of these rare anomalies has risen in recent years, according to various population-based studies due to advances in fetal ultrasound technology.MethodThis retrospective study examines the diagnosis of CLA, and was conducted on 72 patients between March 2014 and March 2024 at Taleghani Pediatric Hospital in Gorgan, Iran.ResultThe average age was 18.8 ± 30.3 months, with the majority being boys (62.5%). Most participants had CCAM (41.7%), followed by CLE (18.1%), BPS (16.7%), pulmonary hypoplasia (9.7%), BC (8.3%), and hybrid lesion (5.6%). The majority of patients were Fars (62.5%), and the average hospitalization days was 9.4 ± 4.5 days. Cardiac anomalies were observed in 19.4% of the patients. 62 patients (86.1%) exhibited respiratory symptoms, and prenatal screening during pregnancy led to the diagnosis in 51 patients (70.8%). Most patients had left lung anomalies (43; 59.7%), and the majority (90.3%) survived. There is a statistically significant relation between needed for surgical treatment and patients’ type of pulmonary lesions (p-value: 0.02). In addition, there was a significant relation between the Fars ethnicity and the presence of cardiac anomalies (p-value: 0.04).ConclusionSome CLAs remain undiagnosed or untreated due to the rare nature of congenital lung anomalies. Nevertheless, improvements in ultrasound and other imaging methods will make diagnosing and managing these anomalies during the prenatal period more prevalent, resulting in enhanced understanding.

흉부 CT 영상에서 심층 감독 및 하이브리드 병변 초점 손실 함수를 활용한 폐암 분할 개선

흉부 CT 영상에서 심층 감독 및 하이브리드 병변 초점 손실 함수를 활용한 폐암 분할 개선

A Large Mandibular Anterior Swelling—A Diagnostic Dilemma: A Case Report

AbstractHybrid lesions are lesions that combine characteristics of many pathologies. The literature has shown a rare relationship between aneurysmal bone cysts, fibro-osseous lesions, and a central giant cell lesion. In this case, we describe an unusual hybrid lesion in the mandibular anterior area. As a result, the goal of this article is to highlight the significance of comprehensive clinical, radiographic, and histological investigation in addition to presenting a rare instance of hybrid lesion. This will lessen the likelihood of misdiagnosis in these situations and aid in prompt treatment planning of the lesion.

Imaging and Histological Characteristics of Non-Hybrid and Hybrid Fibro-Osseous Lesion at Maxillofacial Area

Background: The integration of clinical, radiographic and pathology findings-based information has long been intrinsic to diagnosis and proper management. Objective: The purpose of study was to observe the imaging features of hybrid and non-hybrid Fibro-Osseous Lesion at Maxillofacial Area among the participants. Methodology: This cross-sectional study was conducted in the Department of Oral &amp; Maxillofacial Surgery at Dhaka Dental College &amp; Hospital, Dhaka, Shaheed Suhrawardy Hospital, Dhaka and Bangabandhu Sheikh Mujib Medical University, Dhaka, Bangladesh from January 2004 to December 2005. Participants were selected for the study who attended into outpatient and inpatient departments with fibro-osseous lesions during study period, irrespective of age &amp; sex. A standardized structured data collection sheet was used to collect necessary information of the study history of the patients and clinical examination; radiology, histopathology and macroscopic findings of surgical specimen were recorded. Result: Most of the patients’ age was between 11 to 30 for both hybrid and non-hybrid form. Among them average age of the patients was 24.98 years (±SD 13.4).The mean age of the Non-hybrid group is 26.35 (±SD 13.92) years and mean age of Hybrid group is 17.17 (± SD 6.18) years. In hybrid form most of the radiological pattern was mixed type (83.3%) but in non-hybrid form all other mentioned radiological character were found. In cross-section of surgical specimens of hybrid form cavitation were found in all cases (100.0%) but it was not detected in a single case in non-hybrid form (0.0%). Multicystic haemorrhagic spaces were seen in 100.0% cases of hybrid form whereas none was detected in non-hybrid. Conclusion: The Imaging and histomorphology of fibro-osseous lesion is very important tools to proper diagnosis and management. Journal of Current and Advance Medical Research, July 2022;9(2):79-82

A Hybrid Lesion of the Mandible with Three Co-existent Pathologies: Report of a Rare Case with Literature Review

Hybrid lesions are rare and microscopically composed of areas of distinct pathologic entities. The occurrence of dual lesions containing Central Giant Cell Lesion (CGCL) and fibro-osseous lesions has been recorded in the literature. Although many similar cases have been reported the pathophysiology of such lesions is yet not well understood. Furthermore, their clinical behaviour and treatment guidelines are also not clearly outlined. Presented here is a unique case of a 17-years old male with a hybrid lesion with a triad of pathological entities, Ossifying fibroma, CGCL and aneurysmal bone cyst. The clinical presentation, pathogenesis and behaviour of the reported lesion are comprehensively explained. The authors believe that greater knowledge of the hybrid variety of jaw lesions may aid in better understanding and management of these lesions by clinicians.

Juvenile psammomatoid ossifying fibroma with aneurysmal bone cyst in the mandible

Aneurysmal bone cysts (ABCs) are rare benign lesions seen as locally destructive, rapidly expansile, and mostly affecting the long bones and vertebrae. The association of ABCs with juvenile psammomatoid ossifying fibroma (PsJOF) is predominantly seen in the extra gnathic region, and it is extremely rare with only a few cases reported so far in the mandible. Here, we report one such case of a hybrid lesion in a 30-year-old male, who presented with a solitary swelling of the right mandible showing partial obliteration of lingual vestibular sulcus, which was histologically confirmed as PsJOF as a preexisting lesion, transforming into an ABC. Such hybrid lesions are usually misdiagnosed and have been sparsely reported in the literature.

Vascular Anatomy in Congenital Lung Lesions-Description and Classification.

BackgroundBronchopulmonary sequestration (BPS) and hybrid lesion of congenital pulmonary airway malformation (CPAM) are congenital lung lesions typically presenting with systemic vascular connection. We describe and categorize this atypical systemic vascular anatomy in congenital lung lesions.MethodsIn a medical chart review from 2005 to 2020 patients with systemic vascular connection of congenital lung lesions were identified. Clinical and radiological data were collected and compared. Two experienced pediatric radiologists reviewed postnatal thoracic contrast-enhanced computed tomography scans to describe and categorize atypical vascular anatomy. We completed our findings with a review on vascular anatomy in congenital lung lesions.ResultsA total of 21 patients with congenital lung lesions (nine extralobar BPS, five intralobar BPS, seven hybrid lesions) had systemic arterial supply; with seven of these additionally having systemic venous drainage. Origin of the feeding arteries from the aorta or aortic main branches was described as supra-diaphragmatic (descending thoracic aorta) in nine and infra-diaphragmatic in ten patients (abdominal aorta, celiac trunk). In two patients with hybrid lesions both supra- and infra-diaphragmatic arterial feeders were found. Additional systemic venous connection of supra-diaphragmatic type drains into the azygos-hemiazygos system (4/21) while the infra-diaphragmatic type (3/21) drains into caval vein, portal or splenic vein.ConclusionVarious variants of systemic arterial and venous connection of congenital lung lesions can be found. Classification of systemic arterial connection as well as venous drainage of congenital lung lesions as supra-diaphragmatic and infra-diaphragmatic types is intuitive, simple and may be important for the surgeon to avoid unanticipated situations and to perform safe resections.

PERIPHERAL KERATOAMELOBLASTOMA/SOLID KERATOCYSTIC ODONTOGENIC TUMOR

<h3>Introduction</h3> Keratoameloblastoma (KAB) is considered a rare variant of ameloblastoma (AMEL). Three histomorphologic types exist: a) simple (acanthomatous ameloblastoma), b) complex with features of both AMEL and keratocyst odontogenic tumor (KOT/OKC), and c) papilliferous revealing papillary keratin-containing extensions in cystic ameloblastomatous lumina. There are two opposing theories on the development of KAB; 1) the tumor arises de novo as an ameloblastoma with exuberant keratin production, and 2) ameloblastomatous transformation occurs in preexisting KOT/OKC. Rare reports exist of solid KOT/OKC (SKOT) sharing features with KAB suggesting a hybrid lesion. Recently, it has been suggested that KAB should be reclassified as SKOT based on the presence of <i>ptch1</i>variants. Herein, we present a peripheral odontogenic tumor with features of KAB and SKOT. <h3>Materials and Methods</h3> 54-year-old male presented with a firm tender swelling of the right side of maxilla in the area of the premolars causing saucerization of the alveolar bone. <h3>Results</h3> Originating from surface epithelium, the tumor exhibited plexiform, follicular and cystic growth patterns with features encountered in KOT/OKC and stellate reticulum areas and reverse polarization in the periphery. Foci of mild pleomorphism and a few scattered mitoses were noted. Plexiform and cystic areas featured abundant keratin. Elongated lingulae infiltrating the connective tissue as well as plexiform extensions with predominantly ameloblastomatous features and few areas of keratinization were appreciated on the surface epithelium. The stroma revealed plasmacytic inflammation and multiple calcifications representing calcifying keratin deriving from burst cysts. Cytokeratin 19 plasmalemmal staining was observed. Keratin in calcifications was also stained. Ber-EP4 revealed focal plasmalemmal reaction in the surface epithelium with ameloblastomatous changes. There was no staining for calretinin and BRAF V600E. Variable Ki67 nuclear staining was noted. <h3>Conclusions</h3> Features of KAB and SKOT were observed. Next generation sequencing for the presence of <i>braf</i> or <i>ptch1</i> variants is underway.

A Literature Review of the Rare Coexistence of Central Giant Cell Granuloma with Aneurysmal Bone Cyst: A Case Report.

Introduction:Central giant cell granuloma (CGCG) is a benign bone tumor that occurs more in young females and anterior of the mandible. It can be unilocular or multilocular with wispy-septation, undulating borders, cortical expansion, and perforation. Central giant cell granuloma in association with other benign lesions of the jaws is named hybrid lesion. An aneurysmal bone cyst (ABC) is a rare, rapidly growing benign tumor that is commonly developed in young females and the mandible molar and ramus regions. It is usually a well-defined cyst-like expansile lesion with an internal structure similar to CGC lesions in radiographic features.Case Report:A 17-year-old girl was referred to the radiology department for panoramic radiography at the end of orthodontic treatment. The complete opacification of the right maxillary sinus, root resorption, and periodontal ligament widening was evident in panoramic radiography. Cone-beam computed tomography revealed a soft-tissue mass and displacement of the lateral nasal wall. The lesion was multilocular with wispy septation and ground glass in some parts. On T2-weighted magnetic resonance imaging, a heterogeneous mass with low to intermediate signals and fluid-fluid levels were observed. The patient underwent surgical curettage, and the histopathological diagnosis was the coexistence of CGCG and ABC.Conclusion:An unusual view of the coexistence of CGCG and ABC could be a lesion with ground glass pattern calcification. Hybrid lesions with the coexistence of CGCG and ABC are rare, and only six cases are reported in the literature in this regard.

A Rare Pediatric Case Report of a Hybrid Lesion of Congenital Pulmonary Airway Malformation and Bronchopulmonary Sequestration

Congenital malformations of the lungs are rare. Congenital pulmonary airway malformation and bronchopulmonary sequestrations are commoner malformations amongst them. Co-existence of both in a single lesion is extremely rare. Such hybrid lesions or rather malformations have been sparsely reported. We report a similar lesion in an eleven years old male who presented with hemoptysis and diagnosed to have a hybrid lesion.

Characterization of the Hyperintense Bronchus Sign as a Fetal MRI Marker of Airway Obstruction.

Background Fetal MRI-based differential diagnosis of congenital lung malformations is difficult because of the paucity of well-described imaging markers. Purpose To characterize the hyperintense bronchus sign (HBS) in in vivo fetal MRI of congenital lung malformation cases. Materials and Methods In this retrospective two-center study, fetal MRI scans obtained in fetuses with congenital lung malformations at US (January 2002 to September 2018) were reviewed for the HBS, a tubular or branching hyperintense structure within a lung lesion on T2-weighted images. The frequency of the HBS and respective gestational ages in weeks and days were analyzed. Areas under the curve (AUCs), 95% CIs, and P values of the HBS regarding airway obstruction, as found in histopathologic and postnatal CT findings as the reference standards, were calculated for different gestational ages. Results A total of 177 fetuses with congenital lung malformations (95 male fetuses) and 248 fetal MRI scans obtained at a median gestational age of 25.6 weeks (interquartile range, 8.9 weeks) were included. The HBS was found in 79% (53 of 67) of fetuses with bronchial atresia, 71% (39 of 55) with bronchopulmonary sequestration (BPS), 43% (three of seven) with hybrid lesion, 15% (six of 40) with congenital cystic adenomatoid malformation, and 13% (one of eight) with bronchogenic cyst at a median gestational age of 24.9 weeks (interquartile range, 9.7 weeks). HBS on MRI scans at any gestational age had an AUC of 0.76 (95% CI: 0.70, 0.83; P = .04) for the presence of isolated or BPS-associated airway obstruction at histopathologic analysis and postnatal CT. The AUC of HBS on fetal MRI scans obtained until gestational age of 26 weeks (AUC, 0.83; 95% CI: 0.75, 0.91; P < .001) was significantly higher (P = .045) than that for fetal MRI scans obtained after gestational age 26 weeks (AUC, 0.69; 95% CI: 0.57, 0.80; P = .004). Conclusion The hyperintense bronchus sign is a frequently detectable feature at fetal MRI and is associated with airway obstruction particularly before gestational age 26 weeks. © RSNA, 2021 Online supplemental material is available for this article. See also the editorial by Dubinsky in this issue.

Automated Diagnostic Hybrid Lesion Detection System for Diabetic Retinopathy Abnormalities

Background: Early diagnosis, monitoring disease progression, and timely treatment of Diabetic Retinopathy (DR) abnormalities can efficiently prevent visual loss. A prediction system for the early intervention and prevention of eye diseases is important. The contrast of raw fundus image is also a hindrance in effective manual lesion detection technique. Methods: In this research paper, an automated lesion detection diagnostic scheme has been proposed for early detection of retinal abnormalities of red and yellow pathological lesions. The algorithm of the proposed Hybrid Lesion Detection (HLD) includes retinal image pre-processing, blood vessel extraction, optical disc localization and detection stages for detecting the presence of diabetic retinopathy lesions. Automated diagnostic systems assist the ophthalmologists practice manual lesion detection techniques which are tedious and time-consuming. Detailed statistical analysis is performed on the extracted shape, intensity and GLCM features and the optimal features are selected to classify DR abnormalities. Exhaustive statistical investigation of the proposed approach using visual and empirical analysis resulted in 31 significant features. Results: The results show that the HLD approach achieved good classification results in terms of three statistical indices: accuracy, 98.9%; sensitivity, 97.8%; and specificity, 100% with significantly less complexity. Conclusion: The proposed technique with optimal features demonstrates improvement in accuracy as compared to state of the art techniques using the same database.

Interstitial Laser Occlusion of the Systemic Feeding Vessel in a Hybrid Lung Lesion: Technique, Clinical Course, Perinatal Outcome and a Review of Literature

AbstractThe two most common fetal lung lesions include Bronchopulmonary sequestration (BPS) and Congenital pulmonary airway malformation (CPAM).Their co-existence is known as a hybrid lesion. Fetal hydrops in such cases is associated with very high morbidity and mortality. Several fetal interventions have been recommended over the past years to treat the same, with varying success rates. We describe a case of a hybrid CPAM complicated by hydrops which was successfully treated by interstitial laser coagulation of the systemic feeder artery. We also review current literature on the available interventions and conclude that interstitial laser has emerged as the preferred modality of treatment over the years.

Desmoplastic Ameloblastoma with Prominent Osteoplasia (Osteoplastic Ameloblastoma) as a Recurrence after 14 Years: A Case Report and Literature Review

Background: Ameloblastoma is the most common odontogenic tumor arising from the odontogenic epithelium and is known for its distinct aggressive clinical behaviour and characteristic histologic picture. Very few cases of desmoplastic ameloblastoma with prominent osteoplasia (osteoplastic ameloblastoma) exhibiting formation of metaplastic bone trabeculae rimmed by active osteoblasts have been described. Case Presentation: We report an interesting case of desmoplastic ameloblastoma with prominent osteoplasia (osteoplastic ameloblastoma) in a 38-year-old male presented as a recurrence after 14 years in the anterior mandible. Conclusion: A hybrid lesion of desmoplastic ameloblastoma with osteoplastic pattern (osteoplastic ameloblastoma) needs inclusion of new cases to understand their behaviour. Recurrence of lesion after 14 years of initial surgery in our case presents the importance of regular bi-annual follow-up for lifetime.

Hybrid Lesion of Ameloblastoma and Adenomatoid Odontogenic Tumour (AOT): Report of Two Cases from a Tertiary Referral Hospital in Sub-Saharan Africa

Introduction: Hybrid lesions are lesions showing the combined histopathological characteristics of two or more previously recognized odontogenic tumours and /cysts of different categories. Hybrid lesions do exist because of close interrelationship of several odontogenic lesions and also because odontogenic tumors and cysts can arise at any stage of odontogenesis. The objective of this study was to present 2 cases of hybrid odontogenic tumour that is composed of adenomatoid odontogenic tumour (AOT) and ameloblastoma.&#x0D; Case Reports: &#x0D; Case 1: A 33year old female patient with a bucco-lingual swelling in the left mandibular premolar-molar-ramus regions of 13years duration. The lesion measured about 15x5x3cm, it is non tender. Surgical specimen revealed hybrid lesion of granular cell type ameloblastoma and AOT.&#x0D; Case 2: An 18year old female patient with a painless right mandibular molar-ramus swelling of 10years duration. Mandibulectomy specimen measured about 11 cm x 8.5 cm x 5 cm and was found to be hybrid lesion of acanthomatous ameloblastoma and AOT.&#x0D; Conclusion: Both cases had a very long clinical duration and showed more buccal bone expansion with barely noticeable lingual bone expansion. With such clinical scenarios, a suspicion of hybrid tumour should be made.

Structural and perfusion magnetic resonance imaging of congenital lung malformations

BackgroundA radiation-free advanced imaging modality is desirable for investigating congenital thoracic malformations in young children.ObjectiveTo describe magnetic resonance imaging (MRI) findings of congenital bronchopulmonary foregut malformations and investigate the ability of lung MRI for their classification.Materials and methodsThis is a retrospective analysis of consecutive MRI examinations performed for suspected congenital lung anomalies in 39 children (median age: 3.8 months, range: 2 days–15 years). Morphological and perfusion findings were characterised on respiratory-gated fast spin echo and dynamic contrast-enhanced sequences obtained at 1.5 tesla. Abnormalities were classified independently by two readers and compared to an expert diagnosis based on pathology, surgery and/or other imaging.ResultsMain diagnoses included bronchopulmonary lesions in 33 patients, scimitar syndrome in 4 patients, pulmonary arteriovenous malformation and oesophageal duplication cyst in one patient each. Of 46 observed abnormalities, 44 (96%) were classified correctly with very good interobserver agreement (96% concordance rate). The 39 detected lung lesions included isolated overinflation (17/39, 44%), cystic pulmonary airway malformation (8/39, 21%), bronchopulmonary sequestration (7/39, 18%), bronchogenic cyst (4/39, 10%) and hybrid lesion (3/39, 8%). All lung lesions presented as perfusion defect at peak pulmonary enhancement. Non-cystic lesions showed a delayed peak (median delay: 2.8 s, interquartile range: 0.5 to 4.0 s) in relation to normal lung parenchyma.ConclusionA dedicated lung MRI protocol including respiratory compensated sequences, dynamic angiography and perfusion is able to reliably delineate parenchymal and vascular components of congenital bronchopulmonary foregut malformations. Therefore, MRI may be considered for comprehensive postnatal evaluation of congenital thoracic malformations.

Hybrid ameloblastoma and central giant cell lesion: Challenge of early diagnosis

Hybrid lesions encompass the occurrence of different entities in one lesion. A 67-year-old woman was referred to the Oral and Maxillofacial Surgery Service for treatment of mandibular Central Giant Cell Lesion (CGCL) previously diagnosed. Intraoral examination revealed edentulism and a painless swelling extending from the alveolar ridge to the buccal vestibule with hard consistency covered by normal mucosae, with unknown duration. Panoramic radiograph revealed a large, multilocular and well-defined radiolucent lesion extending from the region of left mandibular lateral incisor teeth to right mandibular first molar with no evidence of osseous perforation. Initially, a treatment with intralesional injection of corticosteroids was performed. After 18 months of treatment, an increase in size of the osteolytic lesion was noted. An incisional biopsy was carried out and the microscopic examination revealed a unicystic ameloblastoma associated to CGCL. It was performed marsupialization and later the enucleation of residual lesion. The follow-up remains being performed. Key words:Hybrid lesion, central giant cell lesion, ameloblastoma.

41. Hybrid lesion of the jaws: Central odontogenic fibroma and giant cell granuloma

41. Hybrid lesion of the jaws: Central odontogenic fibroma and giant cell granuloma

Cirugía fetal en tumores pulmonares mediante escleroterapia

Las tumoraciones o masas pulmonares fetales microquísticas, tales como la malformación adenomatoidea quística congénita (CCAM, por sus siglas en inglés) y el secuestro bronco pulmonar (SBP), representan patologías congénitas poco frecuentes, diagnosticadas por ultrasonido durante la evaluación prenatal, con un alto índice de mortalidad de 95% como consecuencia de las complicaciones prenatales por hidropesía y fallecimiento posnatal debido a hipoplasia pulmonar. Las terapias de mínima invasión usando agentes esclerosantes han tomado posicionamiento entre las técnicas de bajo costo y alta efectividad para dichas patologías. Metodología. Se realizó un estudio clínico prospectivo, aleatorio, ciego, en 17 embarazadas con diagnóstico de CCAM microquística o de lesión híbrida (CCAM +SBP) tratadas con betametasona o con polidocanol, optando por la terapia alternativa en caso de fallar la técnica inicial. Resultados. En 4/9 (44,4%) de los 9 casos tratados con betametasona se requirió la terapia alternativa de esclerosis con polidocanol para la resolución del cuadro, que comprometía el estado fetal. La involución de la lesión pulmonar y del cuadro hidrópico fue más rápida y progresiva con la escleroterapia percutánea en comparación con el efecto esteroideo materno. Los resultados perinatales fueron asimismo más satisfactorios en el grupo con polidocanol (91,7%) que en el grupo con el esteroide (60%). Conclusiones. Se comprobó la eficacia de la escleroterapia percutánea con polidocanol para el tratamiento de la CCAM y de lesión híbrida, con resolución más rápida y eficaz de la patología en los casos donde existía resistencia a la terapia esteroidea previamente administrada.