Benign bony lesions of paranasal sinuses and skull base: from osteoma to fibrous dysplasia.

PURPOSE OF REVIEW: Benign bony lesions of the craniofacial complex are relatively common. However, their location close to critical neurovascular structures may render their treatment, if required, highly challenging.This article reviews the current literature on their pathophysiology, diagnosis, natural course and treatment, with a focus on most recent findings. RECENT FINDINGS: A new classification has been suggested concerning endoscopic resectability. The ratio of lateral frontal to interorbital distance can accurately and reliably predict the endoscopic reach to lateral frontal sinus, while orbital transposition can assist us in reaching lateral frontal sinus when anatomy is unfavorable. New and combined endoscopic transnasal and transorbital approaches are now in the surgical armamentarium. Prophylactic optic nerve decompression in fibrous dysplasia is absolutely contraindicated as it leads to worse visual outcomes. Radiotherapy of such lesions is of no benefit and may lead to a higher risk of malignant transformation. The presence of Guanine Nucleotide binding protein Alpha Stimulating (GNAS) mutation in chromosome 20 is universally present in fibrous dysplasia and can differentiate them from ossifying fibromas. SUMMARY: Diagnosis and therapeutic management of benign craniofacial bone lesions remains challenging. If surgical treatment is contemplated, the morbidity of the intervention should always be weighed against the potential benefits. Evolution of extended endoscopic endonasal and transorbital surgery means that more lesions can be reached purely endoscopically with better oncological and cosmetic results.

Subdural osteoma in an adolescent patient with epilepsy: an unusual case report and literature review.

OBJECTIVE: Subdural osteoma (SO) is a rarely reported benign tumor, and there is no report of SO manifested with epileptic seizures. We aim to further the understanding of SO-related epilepsy. METHODS: Here, we report a meaningful case of epilepsy secondary to SO. A systematic review of the literature about SO using the electronic database PubMed and Web of science up to December 2022 was conducted. RESULTS: A 15-year-old girl presented with epileptic seizures for 8 years. Magnetic resonance imaging revealed an irregular lesion with heterogeneous signal in the right frontal convexity. Right frontal craniotomy was performed to remove the lesion. The pathological diagnosis was SO. Histological analysis revealed that the mechanosensitive ion channels Piezo 1/2 were upregulated in the brain tissue compressed by the osteoma, compared with the levels in the osteoma-free region. Seizure freedom was obtained during the 6-month follow-up after the surgery. We identified 24 cases of SO in 23 articles. With our case, a total of 25 cases with 32 SOs was included. Of 25 cases, 24 are adults, and 1 is a child. Seizure has been reported only in our case. Frontal osteoma was found in 76% of the patients. Symptoms were cured in 56% of the patients after surgery. CONCLUSION: Surgery is a safe and effective approach to the treatment of symptomatic osteoma. Mechanical compression on cerebral cortex may be a predisposing factor of the epileptogenesis caused by the SO.

Choroidal Neovascular Membranes in Retinal and Choroidal Tumors: Origins, Mechanisms, and Effects.

Choroidal neovascularizations are historically associated with exudative macular degeneration, nonetheless, they have been observed in nevus, melanoma, osteoma, and hemangioma involving the choroid and retina. This review aimed to elucidate the possible origins of neovascular membranes by examining in vivo and in vitro models compared to real clinical cases. Among the several potential mechanisms examined, particular attention was paid to histologic alterations and molecular cascades. Physical or biochemical resistance to vascular invasion from the choroid offered by Bruch's membrane, the role of fibroblast growth factor 2 and vascular endothelial growth factor, resident or recruited stem-like/progenitor cells, and other angiogenic promoters were taken into account. Even if the exact mechanisms are still partially obscure, experimental models are progressively enhancing our understanding of neovascularization etiology. Choroidal neovascularization (CNV) over melanoma, osteoma, and other tumors is not rare and is not contraindicative of malignancy as previously believed. In addition, CNV may represent a late complication of either benign or malignant choroidal tumors, stressing the importance of a long follow-up.

Anatomical study of a human skull with multiple osteomas in a seventeenth-century Dutch still-life painting: bone morphology and artistic intention.

Skulls were frequently depicted in seventeenth-century Dutch still-life paintings. Skulls were interpreted as symbols of vanitas-meaning the evanescence of life-but their morphological features have received little attention. This study analyzed a skull with abnormal tumors in a seventeenth-century Dutch still-life painting by a renowned artist, Edwaert Collier (ca. 1642-1708), from anatomical, forensic, and pathological perspectives. The morphology of the cranium and teeth indicated that the skull likely belonged to a middle-aged female. We carefully diagnosed the abnormal masses as multiple osteomas on the skull and left femur, based on clinical studies and paleopathological literature, which reported lesions with a similar appearance to those observed in Collier's work. Furthermore, detailed observations of the cranial sutures and epiphyses of the long bones in his paintings revealed that the artist may have selected bones with a morphology that was suitable for the subject of vanitas. Collier repeatedly depicted the skull with metopism, the rare condition of having a persistent metopic suture in adulthood. A skull with a metopic suture is called Kreuzschädel, meaning the cross skull, because it forms a cruciform by connecting with the sagittal and coronal sutures. The artist might have chosen skulls with metopic sutures, which is reminiscent of the crucifixion of Christ, as an appropriate motif for the vanitas painting. This paper argues that anatomical analysis could explain the hidden meaning of the painting and disclose the fascinating collaborations between anatomy and art in the seventeenth-century Dutch Republic.

The role of choriocapillaris vessel density in the pathogenesis of macular neovascularization associated with choroidal osteoma.

PURPOSE: To detect the vessel density of choriocapillaris (CC) vascular network in eyes affected by choroidal osteoma and in eyes complicated by macular neovascularization (MNV), using optical coherence tomography angiography (OCTA). METHODS: In this retrospective study, twenty-eight eyes of 28 patients were divided into three groups: group 1 including patients with calcified choroidal osteoma, group 2 including patients with decalcified choroidal osteoma, and group 3 including patients with decalcified choroidal osteoma complicated by MNV. OCTA analyzed the vessel density of CC in these lesions localized in the peripapillary region. RESULTS: We enrolled 12 eyes with calcified choroidal osteoma, 11 eyes with decalcified choroidal osteoma, and 5 eyes with decalcified choroidal osteoma complicated by MNV. The eyes with decalcified choroidal osteoma and MNV revealed a statistically significant reduction in vessel density of the CC respect to the other groups (p < 0.001). Moreover, the vessel density of CC in decalcified choroidal osteoma was significantly reduced compared to calcified choroidal osteoma (p < 0.001). CONCLUSIONS: OCTA allowed a quantitative evaluation of choriocapillaris vessel density in choroidal osteoma, in order to detect the changes of this vascular network, which could lead to the development of MNV. Therefore, OCTA could be a new diagnostic tool in the clinical management of the choroidal osteoma. CLINICAL TRIAL REGISTRATION: ClinicalTrials.gov Identifier: NCT05342324.

Osteoma of the zygomatic bone from 16th century Sardinia (Italy) in the framework of a paleoepidemiological study.

OBJECTIVE: The aim of this paper is to evaluate a case of osteoma of the zygomatic bone in a post-medieval individual in the context of a paleoepidemiological approach. MATERIALS: Forty-five skulls from the plague cemetery of Alghero (Sardinia, Italy), dating back to the end of the 16th century, were evaluated for the presence of osteomata on the outer surface of the cranial vault and facial bones. METHODS: Macroscopic examination was performed using standard anthropological methods. The bone presenting the lesion was submitted to radiological examination through cone beam; a 3D reconstruction was obtained with a surface 3D scanner. RESULTS: Only one case of osteoma was observed in an adolescent aged 13-15 years, located on the right zygomatic bone. The lesion consisted of a rounded mass of 0.5 cm in diameter composed of compact bone, as also the radiographic study demonstrates. The prevalence of osteoma on the outer surface of the cranial vault and facial bones in the population of Alghero was 2.2 %. CONCLUSIONS: This case offers the opportunity to enrich the knowledge about the presence of benign tumors among past populations and to evaluate this lesion in dry bone. SIGNIFICANCE: This study provides paleoepidemiological data on the scarce area of benign tumors in paleo-oncology and presents the first documented osteoma of the zygomatic bone in paleopathology. LIMITATION: Histological study was not performed for conservative issues. SUGGESTIONS FOR FUTURE RESEARCH: Increasing the attention to benign tumors is essential to deepen our knowledge about paleoepidemiology of neoplastic lesions.

Retinal Vascular Patterns and Capillary Plexus Reflectivity of Intraocular Tumors; an Optical Coherence Tomography Angiography Study.

PURPOSE: To define the vascular characteristics of malignant and benign intraocular tumors by optical coherence tomography angiography (OCTA) and consequently to determine the advantages and disadvantages of the imaging technique in clinical practice. METHODS: This prospective, descriptive study consisted of choroidal hemangioma, choroidal nevus, choroidal melanoma, ocular melanocytoma, congenital hypertrophy of retinal pigment epithelium (CHRPE), and choroidal osteoma. Retinal angiography images were taken by OCTA in a 6 × 6 mm HD scan protocol. OCTA characteristics were defined by considering different tumor types separately. RESULTS: This study included 93 eyes of 90 patients diagnosed with benign or malignant intraocular tumors. The mean age of the patients was 48.9 ± 16.9 years in the hemangioma group, 55.3 ± 12.9 years in the nevus group, 48.2 ± 13.4 years in the melanoma group, 48 ± 18.9 years in the melanocytoma group, 45 ± 22.9 years in the CHRPE group, and 27.8 ± 7.8 years in the osteoma group. We showed four vascular patterns at the level of the choriocapillaris layer in circumscribed choroidal hemangiomas - the appearance of a "bag of worms," club-like appearance, giant choroidal vessels and normal choriocapillaris. The rates of these vascular patterns were 40%, 30%, 10%, and 20% in treatment-naïve hemangiomas and were 46.1%, 30.8%, 7.7%, and 15.4% in photodynamic therapy-treated hemangiomas, respectively. There was no different vascular structure in the tumor associated with the lesion in the nevus group. There were three different patterns at the choriocapillaris level in the melanomas - a vascular network (10%), avascular areas (30%) and vascular loops (60%). There were some atypical and nonspecific vascular changes demonstrated in the CHRPE and osteoma groups and a fine vascular network was observed in the melanocytoma group. CONCLUSION: The different types of intraocular tumors had specific vascular characteristics which were easily demonstrated by the OCTA machine. This imaging technique can be a useful tool to differentiate these intraocular tumors non-invasively.

Lingual Osteoma-A Case Report and Literature Review.

Osteomas are rare benign bony tumors located in the tongue area. In most cases, patients presenting with a lingual osteoma are asymptomatic but may complain of the sensation of having a foreign body. Fewer than 100 cases of lingual osteoma have been reported worldwide. Here, we present 2 cases of tongue base osteoma that were treated with excision under a laryngoscope.

DARK WITHOUT PRESSURE IN A CASE OF CHOROIDAL OSTEOMA.

PURPOSE: To report a novel case of dark without pressure in a patient with a choroidal osteoma. To our knowledge, this association has not been previously reported. METHODS: Observational case report. Review of clinical examination and multi-modal imaging findings in a patient with a choroidal osteoma and dark without pressure. RESULTS: A 21-year-old African American woman with no significant past medical history presented with a large, unilateral, juxtapapillary, subretinal, orange-colored, ovoid-shaped lesion with macular involvement. An overlying area of mottled pigmentary changes, fibrosis, and atrophy were present. Adjacent to and surrounding the osteoma was an annular band of hyperpigmented mid-peripheral retina with a sharply demarcated scalloped border that abruptly changed to normal-appearing peripheral retina. Multi-modal imaging including wide-field fluorescein angiography, optical coherence tomography, and ophthalmic B-scan were performed. The funduscopic and imaging findings were consistent with a diagnosis of choroidal osteoma and dark without pressure. CONCLUSION: The examination and imaging findings in this patient suggest a unique association between two relatively uncommon lesions, choroidal osteoma, and dark without pressure. Although these two lesions may simply be coinciding in the same eye, there may be an association with space-occupying lesions causing a change in photoreceptor structure.

Clinical experience and treatment approaches in sinonasal osteomas from a Tertiary Care Hospital in Turkey.

OBJECTIVE: Osteomas are slow-growing benign osseous tumors that particularly located in the paranasal sinuses (PS). Here, we aimed to define the clinical symptoms and features, diagnostic conditions caused by osteomas located in four different PSs, to evaluate the surgical indications and methods, to compare the factors that can affect the surgical decision, radiological findings, and prognosis. METHODS: The data of patients with paranasal sinus lesions and diagnosed as osteoma according to the radiological imaging, who applied to our clinic between 2010 and 2020, were retrospectively collected and re-evaluated in the light of clinical, radiological, and pathological data. Patients who underwent surgical treatments and were definitively diagnosed as osteoma by pathology were enrolled in this study. RESULTS: We presented the data of 117 patients retrospectively. Most of these cases (n = 77, 65.8%) had an osteoma located in the frontal sinus, while 32 cases (27.4%) had osteoma in the ethmoid, two cases (1.7%) had in sphenoid and six cases (5.1%) had in maxillary sinus. We found that the presence of symptoms, diameter of osteoma, surgical indications including state of sinus drainage, and chronic/recurrent sinusitis influence the choices of physicians in management of frontal sinus osteomas. Moreover, we found a significant correlation between the grading systems defined to classify the frontal sinus osteoma to be operated (r = 0.878, 95% CI: 0.724-0.949, P<0.0001). CONCLUSION: There are several grading systems useful for the choice of surgical approaches, however, the clinical symptoms and surgical indications should not be neglected in the follow-up of patients with frontal sinus osteoma. If there are concerns about the grade of osteoma and the endoscopic approach is considered not to be sufficient for resection, the surgical procedure may be initiated endoscopically, and, if necessary, it can be combined with an external approach.

Peripheral Osteoma of the Zygoma.

ABSTRACT: Osteoma is a benign, slow growing lesion that consists of compact or cancellous bone. Three types of osteomas could be classified: the central osteoma arising from the endosteum, the peripheral osteoma from the periosteum, and the extraskeletal soft tissue osteoma. in the craniofacial region, peripheral osteomas of the zygoma are quite rare. A literature review identified 7 cases of zygomatic arch and 3 cases of zygomatic body. This is the first report of zygomatic osteoma that was endoscopically removed. This report presents a rare case of osteoma of the zygoma and its endoscopic approach. The authors were able to confirm that endo-scopic approach of this zygomatic osteoma was safe and effective surgical choice.

Management of paranasal sinus osteomas: A comprehensive narrative review of the literature and an up-to-date grading system.

BACKGROUND: Surgical excision represents the unequivocal treatment modality for symptomatic paranasal sinus osteomas. However, the optimal surgical approach and the extent of the surgery, as well as the management stance in the case of an asymptomatic tumor, remain controversial. METHODS: The MeSH terms 'Osteoma', 'Nasal Cavity', and 'Paranasal Sinuses' were used to retrieve articles concerning the management of paranasal sinus osteomas that were published in the last 30 years, the vast majority of which comprised case reports of one or two cases. Original articles or large series of more than six cases were prioritized. RESULTS: Our review summarizes previous findings and opinions relevant to the management of symptomatic and asymptomatic paranasal sinus osteomas. The recent shifts in trends of their management are thoroughly discussed. Currently, an extension of the lesion through the anterior frontal sinus wall; an erosion of the posterior wall of the frontal sinus; a far-anterior intraorbital extension; an attachment to the orbital roof beyond the midorbital point; and some patient-specific adverse anatomic variations that may restrict access, are considered strong contraindications to a purely endoscopic approach. On the grounds of this thorough review, a new grading system for frontal and frontoethmoidal osteomas is proposed to allow better conformity to recent advancements and current clinical, research, and educational needs. CONCLUSION: Over the past 30 years, endoscopic techniques have emerged as the new standard of care for favorably located paranasal sinus osteomas. Nonetheless, open approaches remain indispensable for the management of the more perplexing cases of frontal sinus osteomas.

Ethmoidal osteoma in children: Literature review and presentation of a case report / Osteoma etmoidal en niños: revisión de la literatura y presentación de un caso

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Fibro-Osseous Lesions Of The Craniofacial Complex In Horses: 30 Cases (2001-2019).

OBJECTIVE: To describe the presentation, diagnosis, treatment, and outcome for horses with fibro-osseous lesions of the craniofacial complex. STUDY DESIGN: Retrospective multicenter case series. ANIMALS: Thirty horses evaluated for fibro-osseous lesions of the skull from January 1, 2001 through December 31, 2019 in four centers. METHODS: Medical records were reviewed for signalment, clinical presentation, histological and diagnostic imaging findings, treatment instituted, and outcome. Long-term outcome information was obtained by owner questionnaire or the medical record. RESULTS: Diagnoses included ossifying fibroma in 20 of 30 horses, osteoma in eight of 30 horses, and fibrous dysplasia in two of 30 horses. Twelve of 30 lesions were diagnosed in horses <1 year old, and 20 of 30 lesions originated from the rostral mandible. The most common treatment was rostral mandibulectomy. Recurrence was not reported after complete excision. Incomplete excision was confirmed in eight horses (four ossifying fibromas, three osteomas, and one fibrous dysplasia), and follow-up information was available for seven horses. Recurrence occurred in one horse, while six horses had long-term resolution of clinical signs. Prognosis for survival and return to use was excellent in 23 horses with long-term follow-up. CONCLUSION: Fibro-osseous lesions were uncommon in this multicenter study; they were most commonly diagnosed in young animals and most frequently affected the rostral mandible. Long-term survival was excellent. CLINICAL SIGNIFICANCE: The definitive diagnosis of fibro-osseous lesions of the craniofacial complex in horses is made from results of histopathology and cannot be determined on the basis of clinical presentation alone. Surgical excision is indicated, and prognosis can be favorable even when complete surgical margins are not obtained.

Focal choroidal excavation and pitchfork sign in choroidal neovascularisation associated with choroidal osteoma.

PURPOSE: To describe a complex case of choroidal osteoma complicated by choroidal neovascularisation and focal choroidal excavation and presence of pitchfork sign. METHODS: A young female patient with confirmed diagnosis of choroidal osteoma was followed up for 5 years. Multimodal imaging including optical coherence tomography and fluorescein angiography (Spectralis HRA + OCT; Heidelberg Engineering, Heidelberg, Germany) was performed. RESULTS: Optical coherence tomography showed the presence of a unilateral choroidal osteoma in the first visit which was later followed by the appearance of focal choroidal excavation. After 3 years, choroidal neovascularisation developed in choroidal osteoma area, with subretinal fluid, subretinal hyper-reflective material, hyper-reflective foci and pitchfork sign. Decalcification of the tumour was not noted in this patient. Treatment with one intravitreal injection of Aflibercept resulted in complete disappearance of subretinal fluid, subretinal hyper-reflective material, pitchfork sign and decreased hyper-reflective foci. CONCLUSION: Inflammatory response can be a propulsive element in the development of focal choroidal excavation and choroidal neovascularisation in choroidal osteoma patients.

FUNDUS AUTOFLUORESCENCE PREDICTING GROWTH OF A CHOROIDAL OSTEOMA IN A 13-YEAR-OLD GIRL.

PURPOSE: To describe multimodal imaging findings, including fundus autofluorescence, in a patient with a growing choroidal osteoma. METHODS: Case report. RESULTS: A 13-year-old girl presented with an asymptomatic lesion in the superonasal macula of her left eye. Vision was correctable to 20/20 in both eyes, and the only significant finding on examination was the yellow flat lesion in the left macula. It measured 0.8-disk areas in size. Ocular coherence tomography showed a space occupying lesion in the choroid that had horizontal hyper-reflective lines consistent with cancellous bone. Re-examination in a year showed enlargement. Fundus autofluorescence showed no abnormalities in either eye. Ultrasonography showed a hyper-reflective lesion with associated shadowing that was consistent with a choroidal osteoma. CONCLUSION: Normal fundus autofluorescence was seen in this young girl with a growing choroidal osteoma. Fundus autofluorescence is a proxy for the health of the retinal pigment epithelium. Normal retinal pigment epithelium is the only factor found to be predictive of future growth of a choroidal osteoma. This noninvasive test may prove useful as a guide to determine frequency of examinations especially in younger patients who might be prone to more rapid growth.

Orbital cellulitis secondary to giant sino-orbital osteoma: A case report.

BACKGROUND: Although osteoma is a common benign tumor of the paranasal sinuses, its orbital extension is not common. Secondary orbital cellulitis has rarely been reported in association with sino-orbital osteoma. CASE: A 30-year-old woman presented with left side proptosis, orbital pain and inflammation. Orbital CT scan showed a well-defined giant osteoma in the superonasal part of the left orbit originating from the left ethmoidal sinus associated with opacity of the ipsilateral ethmoidal sinus and infiltration of orbital soft tissue. After treatment by systemic antibiotics, osteoma was resected with combined external and endoscopic surgery and the patient recovered uneventfully. CONCLUSION: Sino-orbital osteoma may manifest primarily as orbital cellulitis and needs early surgical intervention.

[Tumors affecting the temporomandibular joint - a literature review]. / Tumeurs de l'articulation temporomandibulaire ­ revue de la littérature.

Benign and malign tumors can affect the temporomandibular joint (TMJ) as any other articulation. Nevertheless, TMJ tumors are rare and mostly benign. Their clinical expression is varied including symptomatology similar to TMJ dysfunctional disorders, otologic or neurologic pathologies. In some cases, they remain totally asymptomatic. Hence, diagnosis is difficult since the symptomatology can be misleading with TMJ dysfunctional disorders or otologic disorders wrongly diagnosed. There is thus frequently a long delay between symptoms onset and diagnosis. The great variety of TMJ lesions explains the wide range of possible treatment modalities, mostly based on surgery. We provide here a review of the lesions originating from the TMJ. Tumoral or cystic mandibular lesion affecting the TMJ through local extension will not be discussed. Osteoma, osteoid osteoma, osteoblastoma, chondroma, osteochondroma, chondroblastoma, tenosynovial giant cell tumors, giant cell lesions, non-ossifying fibroma, hemangioma, lipoma or Langerhans cell histiocytosis are all possible diagnosis among the benign tumors found in the TMJ. Pseudotumors include synovial chondromatosis and aneurysmal bone cyst. Finally, malign tumors of the TMJ include mainly sarcomas (osteosarcoma, chondrosarcoma, synovial sarcoma, Ewing sarcoma, and fibrosarcoma), but also multiple myeloma and secondary metastases. We will review the clinical, radiological and histological aspects of each of these lesions. The treatment and the recurrence risk will also be discussed.

Printing a patient-specific instrument guide for skull osteoma management.

BACKGROUND: To surgically remove osteoma and to keep an optimal cosmetic profile would be very challenging. To solve the difficulty, we utilized the three-dimensional (3D) printing technologies in generating a patient-specific instrument guide (PSIG) for the safe removal of a skull bone tumor. METHODS: The preoperational brain computed tomography (CT) provided the digital imaging with thin slices, and then images were reconstructed into a 3D skull model. Based on the model, we designed a PSIG to make landmarks on the osteoma to avoid excessive removal of the skull bone. During the operation, the surgeons could remove the osteoma piece by piece by using the landmark as a reference point. RESULTS: The PSIG was successfully applied to remove an osteoma that measured 60 × 48 × 40 mm over the left frontoparietal skull of a female patient. The 3D CT reconstruction taken both before and 4 months after surgery showed a significant change in the appearance of the osteoma. CONCLUSION: The PSIG was able to guide the surgeon in the safe removal of the skull osteoma, as well as in maintaining the cosmetic skull profile.