Clinicopathologic Case Series Research Articles

Phacolytic Glaucoma Diagnosed by Cytopathology: A Clinicopathologic Case Series

ABSTRACT Purpose The aim of this study was to describe the clinical presentation, histopathologic characteristics, and management of phacolytic glaucoma. Patients and Methods The database at the Florida Lions Ocular Pathology Laboratory was searched for surgical specimens at the Bascom Palmer Eye Institute. Patients with a diagnosis of “phacolytic glaucoma” on cytopathology between the years of 1997 and 2023 were included (n = 15). Patient demographics, anatomic site, laterality, clinical features, and ocular examination findings were obtained from available medical records. Results The diagnosis of phacolytic glaucoma was established by light microscopic examination of cytology specimens in 15 eyes (Right = 8, Left = 6, not specified = 1). Between 1997 and 2023, there were 32 cytopathology cases with a preoperative diagnosis of phacolytic glaucoma. From this group of 15 cases, there were nine males and six females. Mean age was 64.2 years (Range: 39–87). Thirteen samples were from the anterior chamber and two were obtained from the vitreous. All 15 cases (100%) demonstrated histiocytes with engulfed foamy and/or granular presumed lenticular material. CD68 immunohistochemistry was positive within histiocytes in four cases that were stained (100%). Of the available medical records, clinical features, and ocular examination findings included: eye redness, decreased vision, eye pain, anterior chamber inflammation, dislocated or subluxed crystalline lens, cataract, and elevated intraocular pressure (average = 41.3 ± 8.67 mm Hg). Conclusions Phacolytic glaucoma is a rare complication of mature/hypermature cataracts that presents with ocular pain, decreased vision, and anterior chamber inflammation. AC paracentesis with cytopathologic evaluation is a minimally invasive, rapid technique that can aid in the diagnosis and management of this disease.

Pustular mycosis fungoides has a poor outcome: a multicentric clinico-pathological and molecular case series study.

Mycosis fungoides (MF) has usually an indolent course. However, some patients develop a more aggressive disease and few prognostic parameters have been identified. Isolated cases of pustular MF (pMF) suggest an unfavourable prognosis. We aim to describe the clinico-pathological characteristics and prognostic value of pMF. We retrospectively collected data of all cases of MF with histological pustules diagnosed from 2009 to 2020. The outcomes and clinico-pathological characteristics of pMF at diagnosis (pMFD) were compared to those of a cohort of non-pustular MF (NpMF). 33 pMF (including 22 pMFD) and 86 NpMF cases were included. The median age at diagnosis of pMF was 61 years [IQR=50-75]. The median follow-up of pMFD was 32 months [IQR=14-49]. Clinically, 33% of pMF had pustules. Large-cell transformation (LCT) occurred in 17 cases. pMFD were at a significantly more advanced-stage and more showed LCT at diagnosis than NpMF (50% vs 7%, p<0.001 and 23% vs 0%, p<0.001, respectively). In multivariate Cox analysis, the presence of histological pustule at diagnostic was associated with shorter OS in all patients (HR=13.90, CI95%[2.43-79]; p=0.003), and in early-stage patients (HR=11.09, CI95%[1.56-78.82]; p=0.02). In multivariate Fine and Gray model analysis, pMFD was associated with a higher cumulative incidence of LCT (SHR=13.90, CI95% [2.43-79]; p=0.003) in all patients. Median OS after the occurrence of histological pustules during follow-up of all pMF patients was 37 months, with a five-year OS of 25% (CI95% [0.06-0.5]). pMF often follows an aggressive course, with a high risk of LCT and shorter survival, even for early-stage patients. Histological pustules at diagnostic of MF might represent an independent poor prognostic factor, to be confirmed by further studies. Because pustules are not always clinically identified, histological pustules should be mentioned in pathology reports of MF and prompt discussion of a closer follow-up.

Eosinophilic Angiocentric Fibrosis of the Orbit: A Clinicopathologic Review of 6 Novel Cases With Review of the Literature

To describe 6 cases and review the current state of knowledge of eosinophilic angiocentric fibrosis (EAF) involving the orbit. Retrospective clinicopathologic case series and review of the current literature METHODS: Clinical records and histopathologic data of orbit-involving EAF were gathered between 2004 and 2022 from a single academic institution. The patients' presenting clinical symptoms and signs, laboratory data, radiographic studies, and management documentation were collected. Retrospective review identified 6 novel cases, totaling 31 cases of EAF involving the orbit described as of this writing. Fourteen patients were male, and the average age of presentation was 49.8 years (range 25-78 years). Eighteen patients had concurrent sinonasal involvement, whereas 13 had primary orbital involvement. The median duration of symptoms prior to evaluation was 24 months, with nasal symptoms, proptosis, periorbital swelling, and pain being the most common presenting symptoms. The majority of patients underwent surgical debulking, as well as treatment with glucocorticoids and steroid-sparing agents, such as rituximab, with varied results. EAF involving the orbit is uncommon. The histopathologic findings include a perivascular, eosinophil-rich infiltrate and a pauci-inflammatory storiform type of fibrosis concentrated around small vessels. Orbital involvement usually results from local extension from adjacent sinuses, but primary orbital involvement has been described. Surgical debulking and immunosuppressive agents such as rituximab have been shown to stabilize disease.

Plasmacytoid urothelial carcinoma - A clinicopathological case series of an aggressive variant of urothelial cancer.

Many new morphological variants of urothelial carcinoma of urinary bladder have been described in the literature, plasmacytoid/signet ring cell/diffuse variant being one of the rare amongst these. Till date, no case series has been reported from India, describing this variant. We retrospectively analyzed the clinicopathological data of 14 patients diagnosed at our center with plasmacytoid urothelial carcinoma. Seven cases (50%) were pure forms while the remaining 50% of cases had a concurrent conventional urothelial carcinoma component. Immunohistochemistry was performed to rule out other mimickers of this variant. Treatment-related data were available for seven patients, while follow-up was available for nine cases. Overall, plasmacytoid variant of urothelial carcinoma is considered to be an aggressive tumor with poor prognosis.

Bilateral Thalamic Gliomas Harboring Alterations of EGFR and H3K27M: An Integrated Clinicopathological Characteristics Case Series

Bilateral thalamic gliomas (BTGs) are rare central nervous system tumors, and the outcome is usually dismal. BTG often harbors an EGFR mutation; however, a mutation in H3K27M is rare. We described 5 cases of BTGs harboring concomitant alterations of EGFR and H3K27M and retrospectively analyzed the clinicopathological features and prognosis of this rare entity. Clinical data of patients were retrieved, and immunohistochemistry and molecular analyses were performed. In addition, a systematic review of literature was conducted using PubMed. Median patient age was 6 years (range, 3-9 years). The male-to-female ratio was 3:2. Tremors and disturbed speech were the main clinical manifestations. All lesions were located at bilateral thalami, and in 3 of 4 patients, the more significant thalamic lesion was on the left. Two patients harbored insertion mutations in exon 20 of EGFR, 1 missense mutation in exon 7 of EGFR, and 2 EGFR amplifications. After a median overall survival of 8 months, 3 patients died as a result of tumor progression. Concomitant alterations of EGFR and H3K27M might indicate a new subtype of diffuse midline glioma, H3K27M-altered. In addition, EGFR alterations could provide potential molecular therapeutic strategies to improve the dismal prognosis of BTGs. Due to the rarity of these tumors, more cases must be collected to study the pathogenesis, treatment, and clinical outcomes of BTGs with double alteration phenotypes.

Multi-Modal Biomarkers of Repetitive Head Impacts and Traumatic Encephalopathy Syndrome: A Clinicopathological Case Series.

Traumatic encephalopathy syndrome (TES) criteria were developed to aid diagnosis of chronic traumatic encephalopathy (CTE) pathology during life. Interpreting clinical and biomarker findings in patients with TES during life necessitates autopsy-based determination of the neuropathological profile. We report a clinicopathological series of nine patients with previous repetitive head impacts (RHI) classified retrospectively using the recent TES research framework (100% male and white/Caucasian, age at death 49-84) who completed antemortem neuropsychological evaluations, T1-weighted magnetic resonance imaging, diffusion tensor imaging (n = 6), (18)F-fluorodeoxyglucose-positron emission tomography (n = 5), and plasma measurement of neurofilament light (NfL), glial fibrillary acidic protein (GFAP), and total tau (n = 8). Autopsies were performed on all patients. Cognitively, low test scores and longitudinal decline were relatively consistent for memory and executive function. Medial temporal lobe atrophy was observed in all nine patients. Poor white matter integrity was consistently found in the fornix. Glucose hypometabolism was most common in the medial temporal lobe and thalamus. Most patients had elevated plasma GFAP, NfL, and total tau at their initial visit and a subset showed longitudinally increasing concentrations. Neuropathologically, five of the nine patients had CTE pathology (n = 4 "High CTE"/McKee Stage III-IV, n = 1 "Low CTE"/McKee Stage I). Primary neuropathological diagnoses (i.e., the disease considered most responsible for observed symptoms) were frontotemporal lobar degeneration (n = 2 FTLD-TDP, n = 1 FTLD-tau), Alzheimer disease (n = 3), CTE (n = 2), and primary age-related tauopathy (n = 1). In addition, hippocampal sclerosis was a common neuropathological comorbidity (n = 5) and associated with limbic-predominant TDP-43 proteinopathy (n = 4) or FTLD-TDP (n = 1). Memory and executive function decline, limbic system brain changes (atrophy, decreased white matter integrity, hypometabolism), and plasma biomarker alterations are common in RHI and TES but may reflect multiple neuropathologies. In particular, the neuropathological differential for patients with RHI or TES presenting with medial temporal atrophy and memory loss should include limbic TDP-43. Researchers and clinicians should be cautious in attributing cognitive, neuroimaging, or other biomarker changes solely to CTE tau pathology based on previous RHI or a TES diagnosis alone.

Does the Anti-Tau Strategy in Progressive Supranuclear Palsy Need to Be Reconsidered? No.

Does the Anti-Tau Strategy in Progressive Supranuclear Palsy Need to Be Reconsidered? No.

Pleomorphic Fibroma: A Clinicopathologic Case Series With the Review of the Literature.

Pleomorphic fibromas are rare flesh-colored benign neoplasms first described in 1989. Their histopathology is notable for nuclear pleomorphism of spindle cells and multinucleate giant cells but lacking mitoses. The cellular origin of these tumors is unknown. This case series describes an additional 18 lesions with discussion of histopathology and immunohistochemistry. This case series of 18 pleomorphic fibromas uses immunohistochemical staining for CD34, CD68, factor XIIIa, and S-100 and general histopathologic examination under light microscopy to describe the lesions. Immunohistochemical stains for CD34 showed nearly universal positivity of the pleomorphic spindle cells, although some more focally. The pleomorphic cells were negative for CD68, variably positive for factor XIIIa, and universally negative for S-100. All the lesions showed characteristic nuclear pleomorphism with absent mitoses. Collagen thickening was variable, mucin was absent, and perivascular inflammation was present in all lesions. Pleomorphic fibromas are fibrous lesions with benign clinical course and histopathologic findings including nuclear pleomorphism. Immunohistochemical staining characteristics of the lesion, along with unique spindle cells and multinucleate giant cells help to differentiate this from other tumors.

Pulmonary Arterial Thrombosis Is a Novel and Underreported Feature in Severe COVID-19 with Fatal Outcome: Results from a Prospective Single-Centre Clinicopathological Case Series

Background: COVID-19 caused by the novel SARS-CoV-2 has rapidly become pandemic and has an estimated mortality rate of 3·4% of infected individuals However, th

Central Nervous System–Invading Eccrine Gland Carcinoma: A Clinicopathologic Case Series and Literature Review

Eccrine carcinoma involvement of the central nervous system (CNS) is exceedingly rare. The prognosis and response to treatment of this pathology remain poorly characterized. A retrospective case series and literature review were conducted. CNS-invading eccrine carcinoma was diagnosed in 3 patients (2 male and 1 female; age range, 60-79 years), including 2 cases of brain metastases and 1 case of brain-invading skull metastasis with subsequent spinal metastasis. The interval from primary tumor to CNS invasion was 18-51 months. All patients received multimodal therapy following diagnosis of CNS involvement. One patient who harbored a NOTCH1 mutation demonstrated a durable oncologic response after treatment with the immune checkpoint inhibitor pembrolizumab and lived 39 months after CNS invasion. The other 2 patients were discharged to hospice care within 1 month after the diagnosis of eccrine carcinoma brain metastasis. Including this case series, 23 cases of eccrine carcinoma invasion or metastasis to the CNS have been reported, with survival after diagnosis of CNS involvement ranging from a few weeks to 4 years. We present 3 cases of eccrine carcinoma metastatic to the CNS, including the first reported case to our knowledge of eccrine carcinoma treated with immunotherapy. This case, harboring a NOTCH1 mutation, demonstrated the longest durable oncologic response reported in this rare disease. Genomic and molecular testing may play increasingly important roles in the evaluation of these metastases.

Hemolysis-associated hemoglobin cast nephropathy results from a range of clinicopathologic disorders

Intravascular hemolysis is relatively rare but can lead to acute kidney injury (AKI), from increased destruction of erythrocytes and release of free hemoglobin. Since hemolysis and hemoglobinuria are known causes of acute kidney injury we sought to define clinicopathologic findings and outcomes of patients with hemolysis-associated hemoglobin cast nephropathy through a retrospective analysis of 27 cases. The mean patient age was 47 years (range 19-79) and the female-to-male ratio was 1.3:1. All patients presented with AKI with a mean serum creatinine of 8.0 (range 2.9-17.0) mg/dL. Etiologies included autoimmune hemolytic anemia (30%), medication (26%), paroxysmal nocturnal hemoglobinuria (7%), procedural/mechanical causes (7%), transfusion of incompatible blood (4%), toxin ingestion (4%), disseminated intravascular coagulation (4%), and hemoglobinopathy (4%). All biopsies showed acute tubular injury and pigmented, proteinaceous casts characterized by positive hemoglobin immunohistochemistry. After a mean follow-up of nine months (range 0.5-26), the mean serum creatinine was 1.3 (range 0.6-3.3) mg/dL, with 78% of patients returning to normal kidney function. Thus, based on our clinicopathologic case series, hemolysis-associated hemoglobin cast nephropathy is an important entity for clinicians and pathologists to recognize as treatment hinges upon elimination of the pathogenic driver of intravascular hemolysis.

Dense deposit disease: a greatly increased biopsy incidence in India versus the USA.

BackgroundWe present the largest clinicopathologic case series to date of dense deposit disease (DDD) in an Indian population and compare the renal biopsy incidence rate to that seen in a large renal laboratory in USA.MethodsCases of DDD were identified and evaluated from native kidney biopsies reported at Renopath, India and at Arkana Laboratories, in the USA. Renopath receives biopsies from four states, located in the South and Eastern part of India. Arkana Laboratories’ biopsies came from 37 states across the USA.ResultsDuring the study period, there were a total of 25 patients diagnosed with DDD among the 7335 native kidney biopsies at Renopath. Thus, the biopsy incidence rate (cases of DDD/total renal biopsies/year) is 0.0034. By comparison, there were 10 cases of DDD diagnosed among 26 319 native kidney biopsies at Arkana Laboratories during the same time period, with a renal biopsy incidence rate of 0.00038.ConclusionsDDD in this Indian subpopulation has similar clinical and pathologic characteristics when compared to previously reported studies. However, the biopsy incidence rate is about 890% or 8.9 times more common in this subset of the Indian population when compared with a broad cross-section of the US population. In addition to potential genetic factors, environmental conditions and chronic infections likely contribute to the markedly higher biopsy incidence rate. Given the much greater number of patients with DDD in this population, further retrospective and prospective studies would allow more rapid progress in understanding the pathogenesis of DDD and thus potential treatment of patients with DDD.

Clinically aggressive phenotype: A clinicopathological case series of atypical pituitary adenomas

ObjectiveAtypical pituitary adenomas were relatively more malignant lesions defined by WHO criteria. However, not all of them show clinically aggressive behavior. Thus, the current WHO criteria of atypical adenoma didn’t seem to be enough to distinguish clinically aggressive adenoma. Therefore, we would like to identify other clinical factors in a cohort of atypical pituitary adenomas to a better identification of clinical aggressiveness. Patients and MethodsIn order to verify predictors of clinically aggressive phenotype among atypical pituitary adenomas, we retrospectively analyzed the clinical characteristics and therapeutic outcomes of consecutive 49 cases. ResultsTotally, 26 cases were identified as clinically aggressive pituitary adenoma. Clinically aggressive lesions were more likely to be functional (46.2% vs. 17.4%, p = .0388) and be detected in males (65.4% vs. 21.7%, p = .0037). Clinically aggressive adenomas also had higher Ki-67 index [5.0 (5.3)% vs. 4.1 (1.3)%, p = .0011] and presented bigger tumor size [11.83 (11.95) cm3 vs. 5.39 (6.08) cm3, p = .0174]. In multivariate analysis, gender (p = .017), functional status (p = .009) and Ki-67 index (p = .024) were independent predictors of clinical aggressiveness. Further analysis revealed that Ki-67 index of more than 4.45% was associated with worse progression-free survival. ConclusionsGender, functional status, tumor size and Ki-67 index ≥4.45% were associated with clinical aggressiveness. A clinicopathological classification of pituitary adenomas may be useful to determine who should be under closer radiological follow-up or followed multimodal treatment strategy.

Atypical pituitary adenoma: a clinicopathologic case series.

OBJECTIVE In 2004, the WHO classified atypical pituitary adenoma as a distinct adenoma subtype. However, the clinical significance of this distinction remains undetermined. The authors sought to define patient characteristics, tumor features, and treatment outcomes associated with atypical pituitary adenoma. METHODS The authors reviewed records of patients who underwent resection of pituitary adenoma at the University of California, San Francisco, between 2007 and 2014. Per institutional protocol, adenomas exhibiting mitotic activity underwent evaluation for all 3 markers of atypicality (mitotic index, extensive p53 staining, and MIB-1 index ≥ 3%). Statistical analyses were performed using χ2, Fisher's exact test, t-test, log-rank, and logistic regression. RESULTS Between 2007 and 2014, 701 patients underwent resection for pituitary adenoma. Among these patients, 122 adenomas exhibited mitotic activity and therefore were evaluated for all 3 markers of atypicality, with 36 tumors (5%) proving to be atypical. There were 21 female patients (58%) and 15 male patients (42%) in the atypical cohort, and 313 female patients (47%) and 352 male patients (53%) in the nonatypical cohort (p = 0.231). The mean age of patients in the atypical cohort was 37 years (range 10-65 years), which was significantly lower than the mean age of 49 years (range 10-93 years) for patients in the nonatypical cohort (p < 0.001). The most common presenting symptoms for patients with atypical adenomas were headaches (42%) and visual changes (33%). Atypical adenomas were more likely to be functional (78%) than nonatypical adenomas (42%; p < 0.001). Functional atypical adenomas were significantly larger than functional nonatypical adenomas (mean diameter 2.2 vs 1.4 cm; p = 0.009), as were nonfunctional atypical adenomas compared with nonfunctional nonatypical adenomas (mean diameter 3.3 vs 2.3 cm; p = 0.01). Among the entire adenoma cohort, larger presenting tumor size was associated with cavernous sinus invasion (p < 0.001), and subtotal resection was associated with cavernous sinus invasion (p < 0.001) and larger size (p < 0.001) on binomial multivariate regression. The median time until recurrence was 56 months for atypical adenomas, 129 months for functional nonatypical adenomas, and 204 months for nonfunctional nonatypical adenomas (p < 0.001). Functional atypical adenomas recurred more frequently and significantly earlier than functional nonatypical adenomas (p < 0.001). When accounting for extent of resection, cavernous sinus invasion, size, age, sex, and functional subtype, atypicality remained a significant predictor of earlier recurrence among functional adenomas (p = 0.002). CONCLUSIONS When compared with nonatypical pituitary adenomas, atypical adenomas are more likely to present in younger patients at a larger size, are more often hormonally hypersecretory, and are associated with earlier recurrence. These features lend credence to atypical pituitary adenomas being a distinct clinical entity in addition to a discrete pathological diagnosis.

Atypical Pituitary Adenoma: A Clinicopathologic Case Series

Atypical Pituitary Adenoma: A Clinicopathologic Case Series

Atypical Pituitary Adenoma: A Clinicopathologic Case Series

Background: In 2004 the World Health Organization (WHO) classified atypical pituitary adenoma as a distinct adenoma subtype. However, the clinical significance of this distinction remains undetermined. Objective: We sought to better characterize patient characteristics, tumor features, and treatment outcomes associated with atypical pituitary adenoma. Methods: We retrospectively reviewed the records of patients undergoing evaluation at the University of California, San Francisco (UCSF). From 2010–14, 34 tumors demonstrated elevated mitotic index, positive p53 staining, and Ki-67 proliferative index >3%, and were included in our study. Log-rank, Kaplan-Meier, and Fisher’s exact test were used for statistical analyses. Results: There were 21 female patients (62%) and 13 male patients (38%), ranging in age from 10 to 65 years with a median tumor size of 2.5 cm. The majority of tumors (65%) demonstrated local invasion, a feature more often associated with macroadenomas (Fisher’s exact test, p = 0.003). There was a trend toward significance when analyzing gender and tumor size; female patients appeared to present with larger tumors (Fisher’s exact test, p = 0.132). Five patients (15%) suffered tumor recurrence, at a median time of 5 years. We found no statistically significant association between hormone positivity, size, extent of resection, invasion, and recurrence. Conclusion: Clinical distinction between typical and atypical adenoma remains elusive. Our analyses suggest that atypical adenomas may tend to be hormonally positive, more invasive as they get larger, exhibit a female preponderance, with a statistical trend toward women presenting more frequently than men with macroadenomas. Factors associated with recurrence remain unclear.

Morphologic Evolution in Post-Transplant Lymphoproliferative Disorders (PTLD): A Clinicopathologic Case Series

Post-transplant lymphoproliferative disorders (PTLDs) are classified as early PTLDs (E-PTLD), polymorphic PTLDs (P-PTLD), monomorphic PTLDs (M-PTLD) and classical Hodgkin's Lymphoma PTLD (HL). These entities are felt to represent a disease continuum, with a precursor-product relationship, though they are morphologically and clinically distinct. However, this process remains poorly understood, and limited evidence exists to support this hypothesis. We report a series of nine cases extracted from a PTLD database, including both pediatric and adult solid organ transplant recipients, with recurrent disease episodes that evince an apparent evolution in their morphologic categorization between episodes. All patients identified were high risk for PTLD, with multiple identifiable predisposing factors. Presentations varied from isolated lymphadenopathy, to gastrointestinal involvement to pulmonary involvement. Four of the patients were deceased at the time of acquisition, though only one directly from PTLD. Eight of the nine identified patients developed E or P-PTLD lesions that preceded a subsequent M-PTLD or HL lesion at a similar tissue site. Two of the cases had metachronous P and M-PTLD lesions. The six M-PTLD subtypes were variable, including DLBCL, Burkitt, T-cell, and extramedullary plasmacytoma, in addition to the three cases of classical Hodgkin's lymphoma. These cases suggest that E and P-PTLD may act as common precursor lesions to the development of both the M-PTLD variants and HL type lesions, and that the PTLD classification schema represents different stages of a common underlying pathology. DisclosuresOwen:Lundbeck: Honoraria; Gilead: Honoraria; Janssen: Honoraria; Roche: Honoraria.

Papillary meningioma in the dog: A clinicopathological case series study

Papillary meningioma (PM) is one of the most aggressive variants of meningioma in humans and classified as grade III by WHO system. To date, the biological behavior of PM is still not clear in dogs. This study investigated the correlation between histopathological findings of 16 canine PMs and follow up data. Moreover, the expression of doublecortin, E-cadherin, and N-cadherin was investigated by immunohistochemistry. The supratentorial compartment resulted the most common involved. Despite the low grade of histological malignancy, 87.5% of dogs that underwent surgery experienced tumor recurrence. Intratumoral necrosis was observed in a strict correlation with malignancy histological parameter and tumor recurrence. The post-surgery mean survival time was much lower than thus observed in the most common histological subtypes. This data were also confirmed in dogs that received a conservative treatment alone. Tumors with a severe clinical behavior showed a high N-cadherin expression versus a low or absent E-cadherin expression.

Ossifying Fibroma of the Jaws: A Clinicopathological Case Series Study

The aim of this study was to assess the clinical, radiographic and microscopic features of a case series of ossifying fibroma (OF) of the jaws. For the study, all cases with OF diagnosis from the files of the Oral Pathology Laboratory, University of Ribeirão Preto, Ribeirão Preto, SP, Brazil, were reviewed. Clinical data were obtained from the patient files and the radiographic features were evaluated in each case. All cases were reviewed microscopically to confirm the diagnosis. Eight cases were identified, 5 in females and 3 in males. The mean age of the patients was 33.7 years and most lesions (7 cases) occurred in the mandible. Radiographically, all lesions appeared as unilocular images and most of them (5 cases) were of mixed type. The mean size of the tumor was 3.1 cm and 3 cases caused displacement of the involved teeth. Microscopically, all cases showed several bone-like mineralized areas, immersed in the cellular connective tissue. From the 8 cases, 5 underwent surgical excision and 1 patient refused treatment. In the remaining 2 cases, this information was not available. In conclusion, OF occurs more commonly in women in the fourth decade of life, frequently as a mixed radiographic image in the mandible. Coherent differential diagnoses are important to guide the most adequate clinical approach. A correlation between clinical, imaginological and histopathological features is the key to establish the correct diagnosis.

Epibulbar Osseous Choristoma: a Clinicopathological Case Series and Review of the Literature

Choristomas are benign, congenital tumours composed of normal tissue in an abnormal location. Osseous choristomas represent the rarest form of epibulbar choristomas, with now 65 cases reported in the literature. We did a retrospective clinicopathological study of all patients with epibulbar osseous choristoma observed at our institution since 1982 and updated the last review of the literature. Three Caucasian male patients, aged between 3 months and 11 years, were identified. All osseous choristomas were located under the superotemporal bulbar conjunctiva of the right eye. All lesions were managed with surgical excision. Histopathology revealed the presence of lamellar bone in all cases, one of which was associated with a dermolipoma. We report a small rare case series of 3 epibulbar osseous choristomas and did a review of the literature. In one patient, the osteoma was associated with a dermolipoma, corresponding to the fourth reported complex choristoma of this type, in an otherwise normal eye, in the literature.