Osteoblastic Rimming Research Articles

7159 Brown Tumor As Primary Presentation Of Parathyroid Carcinoma In A Young Female

Abstract Disclosure: S.G. Zachariah: None. R. Anand: None. B.Y. Wong: None. H.J. Lee: None. Title Brown Tumor as Primary Presentation of Parathyroid Carcinoma in a Young Female Introduction/Background: Parathyroid carcinoma (PC) is a rare cause of hyperparathyroidism (HPT) accounting for less than 1% of primary HPT cases. The mean age of PC presentation is 62 years. Long-standing HPT can lead to the formation of osteitis fibrosis cystica (brown tumors). In today’s world of advanced screening, they are an increasingly rare presentation of HPT. We present a case of PC presenting as multiple brown tumors in a young woman. Case: A previously healthy 32-year-old woman presented to the emergency department with right shoulder pain after a mechanical fall. MRI revealed multiple osseous lesions of the proximal humerus, glenoid, and scapula, concerning for possible metastatic malignancy. Notable labs: calcium 13.6 mg/dL [8.5 - 10.5 mg/dL], phosphorus 1.7 mg/dL [2.5 - 4.5 mg/dL], alkaline phosphatase 2,369 U/L [< 130 U/L], 25-OH vitamin D 0.8 ng/mL [30 - 60 ng/mL], and parathyroid hormone greater than 2,500 pg/mL [20-80 pg/mL]. Albumin and renal function were normal. CT chest/abdomen/pelvis showed lytic lesions of the left 7th rib, anterior superior iliac spine (with nondisplaced fracture), pelvis, and sacrum. Subsequent pelvic biopsy showed fragmented bony trabeculae with osteoblastic rimming and rare osteoclasts, fragments of fibrous tissue with few plasma cells and hemosiderin laden macrophages, possibly consistent with brown tumor. On parathyroid ultrasound, a 2.2 cm cystic mass was seen along the left common carotid artery. CT neck confirmed the location and also reported C7-T1 spinal lesions. Calcium levels stabilized with IV fluids and 90 mg IV pamidronate. She underwent a left parathyroidectomy and left hemithyroidectomy with central neck dissection. Pathology was consistent with a low-grade parathyroid carcinoma, with a foci of angioinvasion and capsular invasion without lymph node metastases. The Ki-67 index was less than 1%. Post-operatively, the patient was started on calcium and calcitriol supplementation and did not experience hungry bone syndrome. On further discussion, the patient noted that she had a jaw tumor that was previously biopsied and found to be benign. The patient has since been referred for genetic testing. Conclusion/Clinical Lessons: Both PC and its initial presentation with brown tumors are uncommon. Even more rare is the presentation of both in a young, otherwise healthy female. In this context, it is also important to note that brown tumors can mimic osseous metastases and appropriate biochemical, imaging, and histopathological evaluation is needed. The diagnosis of PC under 40 years of age is uncommon and genetic testing is indicated. Syndromes including multiple endocrine neoplasia type 1, familial isolated hyperparathyroidism, and hyperparathyroidism-jaw tumor syndrome are potential causes of inherited PC. Presentation: 6/3/2024

Comparative study of cemento-ossifying fibroma and focal cemento-osseous dysplasia in the jaw – 15 year retrospective inquiry and literature review

BackgroundCemento-ossifying fibroma (COF) and focal cemento-osseous dysplasia (FCOD) are benign neoplasms occurring in the mandible and maxilla; histologically, they both show normal bone replacement by a fibrous stroma containing variable amounts of calcified fragments resembling bone and cementum. FCOD is relatively common and requires observation; COF is rare and its growth over time can lead to deformity in the head and neck region. Surgical removal of COF at an early stage is critical to prevent recurrence and deformity. Therefore, distinguishing COF from FCOD is important for proper further management. Methods and MaterialsTo determine the parameters differentiating COF from FCOD, we retrospectively analyzed 15 COF cases and 27 FCOD cases reviewed between 2005 and 2020 at Temple University Hospital. Demographic, radiographic and histopathologic characteristics were compared by using Student’s t test for continuous variables and Chi-square test for categorical variables. ResultsIn the non-histopathologic spectrum, COF occurred at a younger age (average age-31yo versus FCOD 43 yo). FCOD showed female predilection compared with COF. No significant difference was observed regarding location and radiographic density. However, COF was significantly associated with radiographic cortical expansion. In the histopathologic spectrum, FCOD was more frequently associated with significant intralesional hemorrhage, increased vascularity, osteoblastic rimming of trabeculae, and ginger roots –like bone trabeculae. DiscussionWe demonstrated 7 clinical, radiographic, and pathologic parameters that can help differentiate COF from FCOD. Comprehensive assessment of the distinguishing characteristics is valuable for accurate diagnosis and treatment of these two entities, aiding in minimizing unnecessary procedures for FCOD and averting facial deformities caused by COF.

Layered structure of sialoliths compared with tonsilloliths and antroliths.

The aim of this study was to perform a comparative analysis of the ultrastructural and chemical composition of sialoliths, tonsilloliths, and antroliths and to describe their growth pattern. We obtained 19 specimens from 18 patients and classified the specimens into three groups: sialolith (A), tonsillolith (B), and antrolith (C). The peripheral, middle, and core regions of the specimens were examined in detail by histology, micro-computed tomography (micro- CT), scanning electron microscopy (SEM), energy dispersive X-ray spectroscopy, and transmission electron microscopy (TEM). In the micro-CT, group A showed alternating radiodense and radiolucent layers, while group B had a homogeneous structure. Group C specimens revealed a compact homogeneous structure. Histopathologically, group A showed a laminated, teardrop-shaped, globular structure. Group B demonstrated degrees of immature calcification of organic and inorganic materials. In group C, the lesion was not encapsulated and showed a homogeneous lamellar bone structure. SEM revealed that group A showed distinct three layers: a peripheral multilayer zone, intermediate compact zone, and the central nidus area; groups B and C did not show these layers. The main elemental components of sialoliths were O, C, Ca, N, Cu, P, Zn, Si, Zr, F, Na, and Mg. In group B, a small amount of Fe was found in the peripheral region. Group C had a shorter component list: Ca, C, O, P, F, N, Si, Na, and Mg. TEM analysis of group A showed globular structures undergoing intra-vesicular calcification. In group B, bacteria were present in the middle layer. In the outer layer of the group C antrolith, an osteoblastic rimming was observed. Sialoliths had distinct three layers: a peripheral multilayer zone, an intermediate compact zone and the central nidus area, while the tonsillolith and antrolith specimens lacked distinct layers and a core.

Induction of Osteogenesis After BMP-2 Injection in Rabbit Shoulders: A Novel Strategy for Managing Glenoid Bone Defect Associated with Anterior Shoulder Instability

Background: Bone morphogenic protein-2 (BMP-2) has been shown to promote bone formation. The purpose of this research was to determine if an injectable form of BMP-2 could induce bone formation in anteroinferior glenoid bone defects, which is a critical factor of anterior shoulder instability. The purpose of this study was to investigate bone formation in rabbit shoulders following BMP-2 injection into the anterior capsulolabral complex. We hypothesized that the amount of new bone would be greater in rabbit shoulders injected with BMP-2 than in those injected with normal saline. Methods: An experimental study was conducted on 40 shoulders of 20 mature rabbits. Twenty right shoulders were allocated to four groups according to injection dose (5 µg or 10 µg) and follow-up period after injection (4 or 8 weeks). Twenty left shoulders were injected with normal saline as controls. At 4 or 8 weeks after injection, all rabbits were sacrificed, and bone volumes, densities, and locations of bone formation were evaluated using micro-computed tomography (micro-CT). Histologic analysis was performed on all 40 shoulders. Results: Shoulders injected with BMP-2 showed significantly more bone formation than control shoulders in all treated groups (58.6 ± 60.5 mm3 vs. 25.6 ± 32.6 mm3, p = 0.001). Moreover, bone formation increased with dose and follow-up. Locations of bone formation were the lesser tuberosity, greater tuberosity, and anterior capsulolabral complex. Histologic analysis confirmed the micro-CT-detected bone formation by revealing the presence of activated osteocytes, abundant osteoblastic rimming, and woven bone matrix. Conclusion: The study confirms that BMP-2 injection into the anterior capsulolabral complex of rabbit shoulders promotes new bone formation in adjacent soft tissue areas. Study Design: Controlled laboratory study.

Biological Reconstruction for Metacarpal Osteofibrous Dysplasia Treatment: A Case Report

Background: Osteofibrous dysplasia is a rare benign bone tumor characterized by abnormal differentiation of fibrous tissue in bone and commonly involves the maxilla, mandible, and tibia and affects children less than 10 years old. Osteofibrous dysplasia affecting the metacarpal is a very rare case. This study presents a case report to present biological reconstruction in the management of metacarpal osteofibrous dysplasia. 
 Case presentation: A 48-year-old female presented with swelling and pain in her right hand over 1 year. On examination, palpable mass with immobile and hard consistency on the first metacarpal and distal neurovascular within normal limit. Radiographic examination (X-ray and MRI) supports osteofibrous dysplasia. We performed complete resection of the first metacarpal and used a fibular graft to maintain length, followed by arthroplasty of the first metacarpophalangeal joint and arthrodesis of the first carpometacarpal joint. Pathological results showed a Chinese letter appearance with osteoblastic rimming that confirmed osteofibrous dysplasia. After 6 months of follow-up, there is no recurrence, and the functional outcome is excellent (MSTS score 93,33%).
 Conclusion: Osteofibrous dysplasia on the metacarpal and affected adult is a very rare case. Treatment with complete resection of the metacarpal, using a fibular graft to maintain bone length, and arthroplasty MCP joint will give good results (MSTS score 93,33%). Follow-up for recurrence is necessary.

Osteoid osteomas of the hands and feet: a series of 71 cases.

Osteoid osteomas typically arise in the long bones of extremities. Patients often report pain relieved by NSAIDS, and radiographic findings are often sufficient for diagnosis. However, when involving the hands/feet, these lesions may go unrecognized or misdiagnosed radiographically due to their small size and prominent reactive changes. The clinicopathologic features of this entity involving the hands and feet are not well-described. Our institutional and consultation archives were searched for all cases of pathologically confirmed osteoid osteomas arising in the hands and feet. Clinical data was obtained and recorded. Seventy-one cases (45 males and 26 females, 7 to 64 years; median 23 years) arose in the hands and feet, representing 12% of institutional and 23% of consultation cases. The clinical impression often included neoplastic and inflammatory etiologies. Radiology studies demonstrated a small lytic lesion in all cases (33/33), the majority of which had a tiny focus of central calcification (26/33). Nearly, all cases demonstrated cortical thickening and/or sclerosis and perilesional edema which almost always had an extent two times greater than the size of the nidus. Histologic examination showed circumscribed osteoblastic lesions with formation of variably mineralized woven bone with single layer of osteoblastic rimming. The most common growth pattern of bone was trabecular (n = 34, 48%) followed by combined trabecular and sheet-like (n = 26, 37%) with only 11 (15%) cases presenting with pure sheet-like growth pattern. The majority (n = 57, 80%) showed intra-trabecular vascular stroma. No case showed significant cytology atypia. Follow up was available for 48 cases (1-432 months), and 4 cases recurred. Osteoid osteomas involving the hands and feet follow a similar age and sex distribution as their non-acral counterparts. These lesions often present with a broad differential diagnosis and may initially be confused with chronic osteomyelitis or a reactive process. While the majority of cases have classic morphologic features on histologic exam, a small subset consists solely of sheet-like sclerotic bone. Awareness that this entity may present in the hands and feet will help pathologists, radiologists, and clinicians accurately diagnose these tumors.

Fibrous Dysplasia of the Spine-A Case Involving the Polyostotic Form Isolated to the Thoracolumbar Spine.

Fibrous dysplasia of the spine in a polyostotic form is very rare, with fewer than 36 cases discussed in the literature and there is no such case in Indonesia that has been reported. The aim of this report is to present a case from Indonesia of polyostotic fibrous dysplasia isolated in the spine. We report a case of a 38-year-old Sundanese man with a 1-year history of progressive back pain and weakness of both lower extremities. There was no history leading to infection and no previous trauma. A physical examination revealed kyphoscoliotic deformity, a café au lait spot, tenderness at the thoracolumbar region, and neurological deficits. Laboratory studies were within normal ranges. Plain radiographs showed lytic lesion and kyphoscoliosis. Magnetic resonance imaging showed an endosteal scalloping, infiltrative process, expansion, and destruction in the vertebral bodies from T2 to L5. The findings of an aggressive destructive process was highly suspicious of a malignant process, relying on differential diagnosis and metastases, plasma cell myeloma, bone tumor and chronic infectious spondylitis. Histology revealed an irregularly oriented osteoid without osteoblastic rimming but surrounded by fibroblastic proliferation with a C-shaped sign. Investigations revealed a diagnosis of polyostotic fibrous dysplasia of the thoracolumbar spine in isolation. The patient underwent T5-S1 stabilization and bone grafting. At 1 year postoperative, the patient was asymptomatic; there was no recurrence and minimal neurological deficit with grade II on the modified McCormick scale. A case of the polyostotic form of fibrous dysplasia of the spine in isolation has never been reported in Indonesia. The extreme rarity of this type of presentation can pose a diagnostic dilemma, and in cases isolated to the spine, surgical treatment with posterior stabilization, decompression, and bone grafting gives a good functional outcome.

OSTEOBLASTOMA IN THE MANDIBLE: A RARE PRESENTATION

Osteoblastoma is a rare, benign, bone-forming tumor, characterized by the presence of osteoblasts with active proliferation, larger than 20 mm. The lesion mostly occurs in the second or third decades of life and has a predilection for the posterior mandible of men. This is a report of a 47-year-old indigenous woman exhibiting a large painful solid mass in the vestibular region of the right mandibular body, of unknown evolution time. The radiographic examination showed a large irregular radiopaque mass in the right posterior mandible, superior to the mandibular canal. The excisional biopsy and subsequent histopathologic analysis showed a compact mass of richly cellularized immature bone tissue with prominent osteoblastic rimming, containing loose connective tissue inside medullary spaces. Some of the osseous tissue had a blue-bone appearance. The diagnosis was osteoblastoma. The patient is being carefully monitored, and there is no recurrence after 2 years’ of follow-up. Osteoblastoma is a rare, benign, bone-forming tumor, characterized by the presence of osteoblasts with active proliferation, larger than 20 mm. The lesion mostly occurs in the second or third decades of life and has a predilection for the posterior mandible of men. This is a report of a 47-year-old indigenous woman exhibiting a large painful solid mass in the vestibular region of the right mandibular body, of unknown evolution time. The radiographic examination showed a large irregular radiopaque mass in the right posterior mandible, superior to the mandibular canal. The excisional biopsy and subsequent histopathologic analysis showed a compact mass of richly cellularized immature bone tissue with prominent osteoblastic rimming, containing loose connective tissue inside medullary spaces. Some of the osseous tissue had a blue-bone appearance. The diagnosis was osteoblastoma. The patient is being carefully monitored, and there is no recurrence after 2 years’ of follow-up.

Histomorphometric Analysis of Pre- and Post-Denosumab-Treated Giant Cell Tumor of Bone.

Context. Denosumab is a monoclonal antibody against RANK ligand. Its administration in giant cell tumor of bone (GCTB) cases results in elimination of giant cells and new bone formation. Neoplastic stromal cells of GCTB harbor mutation of histone 3.3 and have pre-osteoblastic properties and thus express SATB2. Objectives. To (1) analyze histological changes in post-denosumab-treated GCTB, (2) analyze expression of H3.3G34W and SATB2 in pre- and post-denosumab-treated samples, and (3) to discuss why changes occur in the expression of not only H3.3G34W but also SATB2. Materials and Methods. Hematoxylin and eosin slides of 19 cases of denosumab-treated GCTB were reviewed. Immunohistochemical stains H3.3G34W and SATB2 were performed. The number of positive mononuclear cells were counted and graded. Results. Complete absence of osteoclast-like giant cells (OCLGCs) was noted in most cases along with a fibro-osseous component merging with peripheral shell of reactive bone. Irregular trabeculae of woven bone and osteoid with focal osteoblastic rimming was seen. Spindle cells were arranged predominantly in fascicular pattern. Morphometric analysis of H3.3G34W showed a mean of 68.8% positive stromal cells in pretreatment and a mean of 26.9% positive stromal cells in posttreated specimens with a statistically significant P value (.001). Mean percentage of SATB2-positive stromal cells in the pre- and posttreatment specimens was 36.46% and 20.8%, respectively. Conclusions. Our study validates that denosumab treatment results in marked reduction of OCLGCs with increased osteoblastic activity. Decreased expression of H3.3G34W in posttreatment may be a result of decreased antigenicity of neoplastic mononuclear cells. No significant change in SATB2 expression was noted.

Spectrum of histological features of Denosumab treated Giant Cell Tumor of Bone; potential pitfalls and diagnostic challenges for pathologists

ObjectivesDenosumab is Receptor Activator of Nuclear factor Kappa-B Ligand (RANKL) inhibitor which is being used in the treatment of locally advanced, recurrent and metastatic Giant Cell Tumor of Bone (GCTB). It causes reduction in monocyte recruitment and Osteoclast-Like Giant Cell (OLGC) formation which limits bone destruction. After Denosumab treatment, GCTB exhibit diverse morphological features which can pose diagnostic challenge.Our aim was to study the spectrum of histologic features seen in Denosumab treated GCTB which could be helpful in establishing correct diagnosis. MethodsWe retrieved and reviewed H&E stained microscopic glass slides of 38 GCTB cases who received Denosumab as neoadjuvant treatment. These cases were treated at different institutes and diagnosed at our institute between January 2017 and October 2019. Morphologic features such as presence of residual OLGC, appearances of mononuclear stromal and bony components were assessed along with other non-specific features. ResultsPatients' median age was 29 years. Male to female ratio was 1.53:1. Femur was the most commonly involved bone. Microscopically, peripheral shell of reactive bone was observed in all cases. In 20 (52.6%) cases, there was complete elimination of OLGC. Mononuclear stromal cells were predominantly bland spindle shaped and arranged in fascicular and storiform patterns. Focal atypia was noted in 3 cases. Bony component manifested as trabeculae of woven bone with osteoblastic rimming and immature trabeculae of unmineralized osteoid with haphazardly present osteoblasts.Spectrum of stromal changes included cystic spaces, foamy macrophages, inflammatory infiltrate, hemangiopericytoma-like (HPC-like) vessels, hyalinization, edematous areas and hemosiderin pigment.The tumors showed areas which resembled other bony and soft tissue lesions such non-ossifying fibroma, fibrous dysplasia, osteoblastoma, sclerosing epithelioid fibrosarcoma and osteosarcoma. ConclusionDenosumab treatment induces a variety of changes in GCTB. Clinical history and knowledge of these features are necessary for excluding differential diagnoses and avoiding misdiagnosis.

Intraoral and extraoral hyperpigmentation and an expansile mandibular lesion

A 19-year-old woman was referred to the Oral and Maxillofacial Pathology Resident Clinic at Northwell Health Long Island Jewish Medical Center with a primary symptom of “dark spots on my tongue and lips.” She initially reported a noncontributory medical and social history and was not taking any medication at that time nor had any allergies to report. On further questioning, the patient revealed a prior diagnosis of a condition involving multiple bones, including her femur, humerus, maxilla, and mandible. This condition caused expansion of these bones and created a greater risk of developing a fracture. Previous radiographs of the humerus and femur were reviewed (Figure 1). At 10 years of age, she underwent mandibular recontouring for mandibular expansion and obvious facial asymmetry. The tissue was submitted for histologic examination, which revealed a benign fibro-osseous lesion consisting of irregularly shaped, lamellar trabeculae with minimal osteoblastic rimming in a fibrous stroma exhibiting clefting around the trabeculae (Figure 2). She also reported multiple pigmented cutaneous macules on her right shoulder, right buttock, and right inner thigh that had not changed in size and were of unknown duration. She reported an incident of vaginal bleeding at the age of 7, which on further evaluation was revealed to be a sequela from an ovarian cyst. A search of our institution’s archives revealed that the histologic examination of the tissue showed an ovarian follicular cyst with luteinization, which is not an age-appropriate finding in a prepubescent 7-year-old girl. No other family member had exhibited these signs or symptoms. Figure 2Histologic presentation of tissue submitted after recontouring of expansile mandibular lesion. A. A low-power (hematoxylin-eosin, original magnification ×40) image of the tissue specimen shows irregular trabeculae of woven bone in a somewhat parallel arrangement within a fibrous connective tissue stroma. There is a marked clefting artifact separating the connective tissue stroma from the bony trabeculae. B. A higher power (hematoxylin-eosin, original magnification ×100) view of the tissue shows the minimal osteoblastic rimming around the trabeculae in scattered areas but absent entirely in others. The fibrous tissue stroma, parallel trabeculae arrangement, and cleft artifact can also be seen at this power. View Large Image Figure Viewer Download Hi-res image Dr Roth is an oral and maxillofacial pathology resident, Department of Dental Medicine, Donald and Barbara Zucker School of Medicine, Hofstra/Northwell Health, New Hyde Park, NY. Dr Kelsch is an associate professor, Donald and Barbara Zucker School of Medicine, Hofstra/Northwell Health, New Hyde Park, NY.

Peripheral ossifying fibroma of the anterior maxillary gingiva

Background: Peripheral ossifying fibroma is a rejuvenation of the reactive gingiva, usually occurring in the anterior maxillary gingiva. The condition is often clinically ambiguous when diagnosed on the basis of gingival hyperplastic lesions such as focal fibrous hyperplasia, peripheral giant cell granuloma, peripheral fibroma and pyogenic granuloma because peripheral ossifying fibroma has a tendency to recur with a ratio of around 20%. The literature on the subject predominantly classifies peripheral osifying fibroma as an epulis type, but it has also been identified as a peripheral mesenchymal tumor presenting similar clinical symptoms to ossified fibrous epulis. Purpose: The purpose of this article is to explain the rare case of peripheral ossifying fibroma in the anterior maxillary gingiva which can be clinically misdiagnosed as reactive gingival hyperplastic lesions. Case: A case report of peripheral ossifying fibroma in the left lateral incisor and canine of the maxillary gingiva in a 26 year-old male. The patient chiefly complained of a painless, slow growing gingival enlargement on the upper left jaw during the previous five years. Clinical examination confirmed it to be a single, hard swelling in the 21-24 region, pale in color and with a rough surface. Case management: The procedure constituted a complete surgical excision of the lesion together with the underlying periosteum curettage intended to prevent recurrence. The histopathologic examination results indicated tissue with squamous epithelial lining, stroma consisting of fibroblasts, and immature trabecula with osteoblastic rimming between collagen tissue without signs of malignancy. Osteoblastic rimming has specific features in histopathologic examination of ossifying fibroma. Conclusion: Peripheral ossifying fibroma is a rare solitary enlargement in the oral cavity frequently misdiagnosed as ossified fibrous epulis. A definitive diagnosis is made by means of histopathologic examination. The condition has a low reccurance rate.

An Analysis of Clinical and Histopathologic Features of Fibrous Dysplasia of the Jaws: A Series of 40 Cases and Review of Literature.

Fibrous dysplasia (FD) is a rare condition commonly involving the jaws. While FD has a typical clinical and histological presentation, considerable variation exists. Moreover, overlap of features with other disorders is possible. This study serves to characterize the features of a large case series of FD of the jaws. With IRB approval, the University of Florida Oral Pathology Biopsy Service archive was retrospectively searched from 1994 to 2015 for cases of FD. Epidemiological data, location, duration, clinical and radiographic appearance, clinical impression and exact microscopic diagnosis were recorded. The average age was 37.3years (range 7-87years) with majority of cases in females (67.5%). The most common ethnicity was Caucasian. Maxillary location was predominant (59%), followed by mandible (38%) and multiple locations (3%). Expansion was reported in 78% of cases. Radiographically, most cases exhibited ground glass opacity, however some presented with a mottled or mixed radiopaque/radiolucent appearance. Histologically, a wide variation in terms of stromal cellularity, presence of osteoblastic rimming, and presence of calcified material mimicking cemento-osseous dysplasia was observed. Clinicians and pathologists should be cognizant of the significant variability in clinical, histopathologic, and radiographic presentation of FD, which may pose a diagnostic challenge.

Displasia cemento-ósea del maxilar: problemas de diagnóstico diferencial a propósito de un caso

Cemento-osseous dysplasia is a benign lesion which affects the jaw bones. It is a frequent incidental finding but no aggressive therapy is necessary. However, it may be confused with more aggressive entities, such as ossifying and cementifying fibroma, which do require treatment. There are no clear-cut histopathological criteria to differentiate between these entities. We present a case of a 13-year old boy who underwent a biopsy of a clinically undiagnosed maxillary lesion. Histopathological analysis revealed moderately cellular fibrous tissue with bone trabeculae with no osteoblastic rimming and clusters of intensely basophilic material corresponding to cementum. The final diagnosis was cemento-osseus dysplasia. The main histopathological features important in the distinction of these entities are discussed.

Garré Osteomyelitis Mimicking Osteosarcoma in A 27-Year-Old Man: Histopathologic Approach for Diagnosing Periostitis Ossificans

We present a case of periostitis ossificans, noted as a fast-growing mass involving the posterior mandible in a 27-year-old-man. Image examination showed a hyperdense area in the mandible. With a provisional diagnosis of osteomyelitis or osteosarcoma, incisional biopsy was performed. Histopathologic analysis revealed reactive bone, surrounded by pleomorphic osteoblasts and osteoid matrix close to the bone formation, mimicking an osteosarcoma. The diagnosis was inconclusive; additional biopsies were performed to rule out osteosarcoma. Subsequent microscopic analysis revealed new trabecular bone formation with osteoblastic rimming, arranged in a parallel pattern, and the intertrabecular space was filled with vascularized connective tissue and inflammatory infiltrate. Analysis of the images and the histopathologic aspects led to the diagnosis of periostitis ossificans. An inflammatory odontogenic cyst was also present, associated with the third molar, adjacent to the bone lesion. The patient underwent antibiotic therapy, and complete healing was achieved after 2 months.

Juvenile ossifying fibroma: an analysis of clinico-pathological features in a case series with a literature review

Juvenile Aggressive Ossifying Fibroma (JAOF) is divided into two entities namely Juvenile Psammomatoid Ossifying Fibroma (JPOF) and Juvenile Trabecular Ossifying Fibroma (JTOF). JAOFs are aggressive but benign tumours that require clinico-pathological correlation to arrive at the definitive diagnosis. We report nine new cases of JPOF and 6 cases of JTOF with a brief review of the literature to aid correct diagnosis of these two different entities. According to the clinico-pathological analysis both JPOF and JTOF had occurred in children and adults, with a mean age of 28.6 years and 21 years at presentation respectively. JPOF showed a male predilection while equal gender distribution was observed in JTOF. Both tumours occurred most often in the mandible. Regarding the histopathological features all tumours were unencapsulated. All cases of JPOF showed presence of psammoma bodies which can be considered as a main diagnostic feature. JTOF showed osteoid and immature woven bone trabeculae. Osteoblastic rimming was commonly observed among JTOF but absent in JPOF. Other histopathological features did not show any striking difference between the two lesions. In conclusion, the term “Juvenile aggressive ossifying fibroma” is a misnomer as it can occur in adults as well. Therefore, the word “Juvenile” should be excluded when renaming the tumour which is a “need of the hour”. Further, the study demonstrates the use of demographic, radiological and histopathological features with clinico-pathological correlation to diagnose and exclude mimics of JPOF and JTOF.

Gamma correction 99mTc-hydroxymethylene diphosphonate pinhole bone scan diagnosis and histopathological verification of trabecular contusion in young rats.

The aim of this rat experiment using gamma correction pinhole bone scan (GCPBS) was two-fold: first, to confirm whether specific unwashed micro Tc-hydroxymethylene diphosphonate (Tc-HDP) uptake occurs in trabecular contusion (TC) and washed out uptake occurs in edema and/or hemorrhage-irritated trabeculae, and, second, to histopathologically identify the tissue in which the Tc-HDP uptake is unwashed. Five young Sprague-Dawley rats were used for the contusion model and one rat was used as a control. Trauma was inflicted on the femoral shaft with a free-falling iron ball. The presence of injury was confirmed by means of Tc-HDP pinhole bone scan and radiography with built-in scales. All rats were carefully killed for histopathologic verification. The size and shape of the unwashed high Tc-HDP uptake in TC were assessed on a 50-fold magnified GCPBS (mGCPBS), and the findings were compared with those of hematoxylin eosin (H&E) stain findings. mGCPBS showed TC with osteoblastic rimming and high unwashed Tc-HDP uptake. H&E stain findings showed osteoblastic rimming. The smallest TC was 0.03 mm in transaxial diameter on both mGCPBS and H&E stain findings. The four shapes of TC were bar-like, round, ovoid, and pinpointed in the longitudinal, oblique, and transaxial sections. The size and shape shown on mGCPBS and H&E stain findings were in good accord, demonstrating that TC was coated with osteoblastic rimming, which is pathognomonic of contusion. This sign was not seen for the control rat. mGCPBS is useful in the diagnosis of TC because osteoblastic rimming, typically stained in the base, is marked with unwashed high Tc-HDP uptake.

Histopathology, Immunohistochemistry, and Electron Microscopic features of a Dacryocystorhinostomy Ostium Cicatrix.

The aim of this study is to report the histopathological, Immunohistochemical, and ultrastructural features of a dacryocystorhinostomy ostium cicatrix. A prospective histopathological study was performed in a tertiary eye care setting. Scarred nasal mucosal tissues obtained during endoscopic revisions of 10 previously failed dacryocystorhinostomies secondary to complete cicatricial closure of the ostia were studied. The tissue specimens were analyzed using hematoxylin and eosin, periodic acid-Schiff staining. Special stains used include Masson's trichrome and Alizarin red. Immunohistochemistry was performed using vimentin, smooth muscle actin, CD3, CD5, and CD20. Specimens were processed for ultrastructural analysis as per standard protocols for transmission electron microscopy. The respiratory epithelial regeneration was noted to be complete. Irregular laying of deeply eosinophilic and hyalinized collagen with intervening fibroblasts was noted. Focal areas of new bone formation were seen within the cicatricial tissue with osteocytes and ongoing osteoblastic rimming. The infiltrates were mixture of both T and B lymphocytes and were positive for CD3, CD5, and CD20 immunostaining. Electron microscopy showed disorganized collagen fibrils with numerous fibroblasts and mononuclear inflammatory infiltrate. Amorphous bony osteoid within a fibrillar background with metabolically active osteoblasts showed a vesicular cytoplasm, hyperplastic proliferating mitochondria, large Golgi apparatus, and dense endoplasmic reticulum. There is new bone formation within the dense connective tissues of a dacryocystorhinostomy cicatrix. This study may provide useful inputs for further basic science studies aimed at better understanding of wound healing in failed dacryocystorhinostomy.

Central Ossifying Fibroma With Aneurysmal Bone Cyst-Like Areas

Aneurysmal bone cyst is a benign intraosseous lesion that can develop as a primary or secondary lesion associated with other bone diseases. A 29-year-old female presented with a 48-month history of asymptomatic mandibular cortical expansion. A panoramic radiograph showed a large, multilocular, radiolucent lesion extending anteroposteriorly from the mesial portion of tooth #45 to the distal root of tooth #47. An incisional biopsy was performed. Microscopic examination of the biopsy specimen revealed a highly cellular stroma composed of proliferating fibroblasts and osteoid material of varying sizes; many blood-filled cystic spaces of varying sizes; multinucleated giant cells; hemosiderin deposits; and many scattered trabeculae of immature bone, with single osteoblastic rimming and focal osteoclastic activity. The final diagnosis was central ossifying fibroma with aneurysmal bone cyst-like areas. Complete excision of the lesion, with wide margins, was performed, and no recurrence was observed during the follow-up period.

Paget's Disease in Mandible: A Rare Occurrence in an Indian Sub-Continent.

Female patient aged 50-year-old reported an extra oral swelling of lower jaw since 7 months. Broadening and widening of lower third of the face, with thick & incompetent lower lip seen [Table/Fig-1]. Swelling was nontender in the anterior alveolar bone with expanded cortical plates & no mobility of tooth felt after palpation [Table/Fig-2]. Orthopantomogram (OPG) showed irregular radiolucent areas with interspersed radio opacities confined to the anterior and body of mandible, hypercementosis of lower anterior teeth and resorption of roots [Table/Fig-3]. CT scan showed enlarged entire mandible with irregular intra medullary lytic lesion suggestive of fibrous dysplasia/ Paget’s disease [Table/Fig-4]. Incisional biopsy showed features of PD. The presence of irregular areas of bone formation with osteoblastic rimming of bone seen with haematoxylin and eosin stain at 10x and 20x magnifications [Table/Fig-5a,b]. The haematoxyphilic reversal lines seen within most of the areas of bone formation with vascular fibrous connective tissue with amount of inflammatory cell infiltrate at 10X and 20X magnifications [Table/Fig-6a,b]. Whole body bone scintigraphy performed using injection of 20m Ci Tc-99m MDP confirming increased tracer uptake in the mandible only [Table/Fig-7]. Mild increase in alkaline phosphatase (173 IU/L) seen. Based on these facts diagnosis of PD rendered and referred to an endocrinologist for medical management. Treatment was started with Tab. Risedronic acid 35mg orally once every day for two months. Posttreatment scintigraphy showed decreased tracer uptake in the mandible suggestive of good response to treatment [Table/Fig-8]. Patient followed up for more than 20 months successfully. Every six months patient asked for further follow up. [Table/Fig-1]: Pre-treatment frontal photograph showing facial asymmetry [Table/Fig-2]: Pre-treatment Intraoral view showing expanded cortical plates [Table/Fig-3]: Orthopantomogram showing irregular radiolucent areas with inter­spersed radio opacities [Table/Fig-4]: Computerised tomography scan of axial view of mandible showing irregular intra medullary lytic lesion [Table/Fig-5a&b]: Haematoxylin and eosin staining of Biopsy specimen at 10X & 20X magnification showing irregular areas of bone formation respectively [Table/Fig-6a&b]: Microscopic picture of biopsy specimen showing haematoxyphilic reversal lines with fibrous vascular connective tissue. At 10X & 20 X magnification respectively [Table/Fig-7]: Pretreatment bone scintigraphy showing increased tracer uptake [Table/Fig-8]: Post-treatment bone scintigraphy showing reduced tracer uptake PD is disorder of the skeleton results in enlarged & deformed bones. PD showed predilection of male-to-female (3:2) ratio. PD is rare in India, China and Malaysia [1]. Some studies suggested an association of Vitamin D deficiency [2] & viral infection inclusion bodies resembling paramyxovirus nucleocapsid particles in Paget’s osteoclasts [3]. It may present as a coincident with hyperparathyroidism. The bone shows ground glass appearance during early osteolytic stage, later during osteoblastic stage bone gives ‘cotton wool ’ appearance on X-ray film [3]. Pulp chambers of tooth may be calcified, loss of lamina dura and hypercementosis of roots seen [4]. On the other hand during lytic phase of PD root resorption may occur associated with loosening of teeth [5]. Diagnosis is based on clinical, radiographic, bone scintigraphy and histologic features. Deformity involving skull or bowing of long bones which may result in fracture and neurological signs like deafness, cranial nerve palsies and even neoplastic transformation should be examined by plain radiographs [6]. The biochemical values of total plasma alkaline phosphatase activity is elevated in 85% of patients with untreated PD [7] but in our case marginal increase seen. Management mainly aimed at symptomatic (pain control) and specific treatment (Calcitonin, Bisphosphonates). Intravenous bisphosphonates pamidronate, oral alendronate, oral residronate [8] normalize high levels of bone alkaline phosphatase. In our patient residronate 30mg/day for 2months was advised followed by whole body bone scan which showed marked decrease uptake of isotope and biochemical values are within normal limits. Residronate appeared to have excellent safety profile and well-tolerated [8]. PD is considered to be very rare in Indian subcontinent. Its typical clinical features when it involves the maxillofacial region should lead to a high degree of suspicion to arrive at a diagnosis with unknown aetiology. Residronate appeared to have excellent results. Physicians should be aware of the potential for hypocalcemia when patients with polyostotic PD and markedly elevated indicators of bone remodelling are initiated with powerful anti-resorptive therapy.