This report intends to compare classic presentations of odontogenic myxoma in contrast to our case. We also suggest a comprehensive evaluation of lesions and strongly advocate against premature treatments before reaching a definitive diagnosis. This report intends to compare classic presentations of odontogenic myxoma in contrast to our case. We also suggest a comprehensive evaluation of lesions and strongly advocate against premature treatments before reaching a definitive diagnosis. It rarely occurs in any bone other than jaws and peripherally. It originates from the mesenchymal portion of the tooth germ. It is the third most common odontogenic tumor and accounts for approximately 3%–6% of all odontogenic tumors. It has variable radiological and clinical presentations which entails thorough and prudent evaluation of the lesion. We intend to report a case of a 38‐year‐old female patient who was referred to our private practice oral and maxillofacial surgery office with chief complaint of a persistent painful lesion and teeth mobility in the anterior region of the mandible which had underwent a faulty treatment plan in an outside clinic previously. On clinical examination, the lesion revealed a diffuse and slight enlargement extending from the left mandibular first premolar to the right mandibular canine anteroposteriorly and slightly buccolingually. Overlying skin on the mental region was normal and devoid of any ulcerations, erythema, and rise in temperature. Intraoral examination did not reveal any breach in the mucosa. On radiological evaluation, orthopantomography (OPG) and cone beam computed tomography (CBCT) revealed a well‐defined, multiloculated expansile osteolytic lesion with few small septa spreading inside the lesion extending from the left mandibular first premolar to the right mandibular canine, crossing the midline. Histological evaluation confirmed the definitive diagnosis of odontogenic myxoma. The preferred surgical approach for this lesion was enucleation and curettage accompanied with peripheral ostectomy with concomitant burnishing of teeth roots. Myxomas are rare benign tumors of mesenchymal origin. They are locally invasive and occur in various tissues, including the cardiac, skeletal, cutaneous, subcutaneous tissue, aponeuroses, genitourinary tract, and skeletal muscles. It is the third most common odontogenic tumor of the jaw and most frequently occurs in the second to fifth decade of life. Women are more commonly affected than men and the mandible is the more commonly affected than maxilla. The radiographic appearances are variable as it can range from a unilocular to a multilocular radiolucency with multiple patterns of loculation, hence rendering a definitive diagnosis challenging. Treatment approaches for odontogenic myxoma remain surgical. However, it can vary from more conservative approaches like enucleation and curettage to radical resection with wide margins. This article reports an unorthodox presentation of odontogenic myxoma on account of its location within the jaw, its invasive trait, and alternating radiological features rendering it a diagnostic challenge.The odontogenic myxoma is a painless, locally invasive but benign odontogenic neoplasm of the jaws which most frequently occurs in the second to fifth decade of life and the average age of occurrence ranges from 23 to 30 years.Diagnosis of odontogenic myxoma is primarily based on histopathological and/or clinical and radiological findings. From a clinical point of view, odontogenic myxoma is characterized by presenting insidious and asymptomatic slow growth and expansion of the jaw bones, but with locally aggressive behavior leading to occasional cortical bone destruction, soft tissue infiltration, and dental disorders including root resorption and displacement.Despite its benign nature, high recurrence rates have been reported, specifically after removal by curettage alone.Here, we discuss a rare case of an odontogenic myxoma in an uncommon location which yielded a diagnostic challenge. Furthermore, we review the available literature, discuss differential diagnosis, and available modalities for treatment of this lesion.22.1A 38‐year‐old female reported to an outside private dental practice office with chief complaint of pain and discomfort on her left anterior mandible region 1 year prior to presentation to our office. Initial orthopantomography (OPG) was obtained and a radiolucent lesion measuring 15 × 10 mm surrounding the roots of mandibular left lateral incisor and left canine was noticed  and cone beam computed tomography (CBCT) were promptly ordered. They depicted a well‐defined, multiloculated expansile osteolytic lesion with few small septa spreading inside the lesion. Expansion of the buccal cortex with areas of cortical destruction and root resorption of mandibular left central incisor, right central, and lateral incisors were also noticed. The lesion measured 31.2 × 21.0 mm extending from the left mandibular first premolar to the right mandibular canine , cherubism, multicystic ameloblastoma, intraosseous hemangioma, aneurysmal bone cyst, odontogenic keratocyst (OKC), metastatic tumor, and osteosarcoma.Histologically, the bulk of odontogenic myxoma is made up of loosely arranged, spindle‐shaped and stellate cells, many of which have long fibrillar processes that tend to intermesh with apparently inactive odontogenic epithelium scattered through the myxoid (mucous) ground substance. The loose stroma tissue is mainly made up of hyaluronic acid and chondroitin sulfate as in normal tissues, but excessive in amounts. The present cells in this stroma do not show evidence of significant neoplastic activity including pleomorphism, prominent nucleoli, or mitotic figures. Growth pattern of this lesion is differentiated from other lesions by the fact that it gradually grows by secretion of ground substance rather than cellular proliferation. The gelatinous consistency of myxoma permits the lesion to permeate through bony trabeculation leaving no clear margin, therefore, making its complete removal substantially difficult.Treatment approaches for odontogenic myxoma remain surgical. However, it can vary from more conservative approaches like enucleation and curettage to radical resection with wide margins of 1.0–1.5 cm. Although medical management of odontogenic myxoma including chemotherapy has been utilized in few recurrence cases, its use is not advocated.Odontogenic myxomas are notorious for their very high recurrence rate, consisting up to 25% following enucleation and curettage alone.4Due to its aggressive nature, odontogenic myxoma, though benign, requires extreme levels of awareness throughout the diagnosis, treatment, and follow‐up phases. This benign odontogenic tumor can present with a wide variety of clinical and radiological depictions. As seen in our case, it presented with a unilocular radiolucency encompassing teeth roots, highly mimicking the clinical behavior of a radicular cyst. The alternating multiloculartiy and unilocularity characteristics of this lesion compels the clinician to consider numerous differential diagnosis which can only be fortified by a thorough histological analysis in order to reach a definitive diagnosis which was unfortunately lacking on the side of our patient's primary dentist, leading to unnecessary mistreatments of three endodontically sound teeth and further expansion of the lesion, leaving a larger residual defect. This report intends to compare classic presentations of odontogenic myxoma in contrast to our case in terms of unorthodox region, the fact that it crossed the midline of the jaw, and altered radiographic appearance, evolving from unilocular to multilocular pattern which occurs very rarely. We also suggest a detailed and comprehensive evaluation of lesions and strongly advocate against premature treatments before reaching a definitive diagnosis. As in our case, if proper management had been done by the patient's primary dentist, three teeth could have been salvaged and not mistakenly treated and further destruction of cortical bone could have been prevented.None declared.MS conducted the surgery. RD was responsible for data collection and writing of the manuscript. RD and MS were responsible for editing and revising of the manuscript. All authors discussed the results and contributed to the final manuscript.Ethical approval by Ethics Committee of Tehran Medical Sciences, Faculty of Dentistry, Islamic Azad University.The patient consents to their clinical findings, photographs, radiographs, and results to be reported in this article. The patient understands that their name or initials will not be mentioned anywhere in this report and due efforts will be made to completely conceal their identity, however, a total anonymity cannot be guaranteed.


Electrification accidents is a serious pathology due to the passage of an electric current through the body. The clinical manifestation is mainly represented by skin lesions with burners of different degrees however all the tissues of the body can be affected. The cerebral manifestation is a rare entity described in the literature. In this paper, we report the case of a patient with an electrocution accident manifested by an extradural hematoma who has not encountered a similar case described in the literature. We therefore present this case which poses a poorly understood pathophysiological problem. •This paper presents a 44 years old man electrician was brought by his coworkers to emergency after he had an accident by electrocution to a high voltage current with direct cranial contact point at the vertex. The neurological examination revealed muscle power was 3/5 on right side, with 2nd and 3rd degree burns on his face and scalp.•A cerebral CT scan was performed objectifying an extradural hematoma of the vertex with no sign of trauma the bone structure was intact. This one was surgically evacuated the follow-up was good at first but he developed septic shock which was the cause of his death.•Cerebral bleeding due to electric shock are a rare entity described in the literature, their pathophysiological mechanism is still poorly understood, it is difficult to determine that these lesions are due to an accident of electric shock in real life or due to a head trauma associated. Electrification accidents are due to direct or indirect contact of the body with a source of electric current with a difference in potential between 2 different points .There are 2 types of electrification by high voltage currents and those by low voltage currents. Injury to the central nervous system due to electrification accidents is a rare occurrence, but nevertheless serious. In this paper we describe a case of accidental electrification by a high voltage current causing brain damage .2A 44 years old man electrician by profession, right-handed, with no particular pathological history. was brought by his coworkers to our emergency department after he had an accident at work by electrocution to a high voltage current with direct cranial contact point at the vertex, causing an initial loss of consciousness without associated head trauma. There is no history suggestive of any mental or physical illness.At admission, his GCS scores were E4V5M6. Neurological examination revealed muscle power was 3/5 on right side, 5/5 on left side, no sensitive deficiency with 2nd and 3rd degree burns on his face and scalp , Fig. 2.A cerebral CT scan was performed at the ER 4 hours after the accident objectifying an extradural hematoma of the vertex more marked on the left associated with a left frontal pneumocephaly there was no sign of trauma the bone structure was intact , Fig. 4.The diagnosis of extradural hematoma was obvious. We hospitalized the patient in the neurosurgical intensive care unit after being conditioned, the patient underwent a preoperative assessment, then was sent for surgical management. The surgery was performed under general anesthesia by our chief resident. After removal the bone flap we discovered an extradural hematoma which was evacuated, the origin of the bleeding was the superior longitudinal sinus there were no fracture in the bone.On the follow-up, there was total recovery from the motor deficiency, conscious; the control CT scan was satisfying . The patThis case has been reported in line with the 2020 SCARE guidelines [3Brain injuries secondary to electrification accidents represent a rare entity in the literature. The major parts of clinical studies concerning the neurological manifestations of electrification accidents are made from post-lightning accidents. These complications can be central or peripheral, onset immediately or delayed, and transient or permanent .The physiopathology mechanisms are linked to the direct or indirect effect of the electric current on any tissue of the organism that it encounters during its passage, knowing that the vessels and nerves offer the least resistance and are therefore the seat of the strongest intensity, from which it is recognized that the path of electrification is preferentially vascular-nervous. In addition to the electrothermal effect, there is a cellular effect which causes alterations in the plasma membrane with increased membrane permeability resulting in the formation of electropores in the membrane phospholipid layer . These p•stage 0: no vascular lesion;•stage 1: small changes in the wall, often at the level of the intima, with edema or even detachment of endothelial cells, aggregation of blood along the wall; these lesions are localized in apparently healthy tissue in the vicinity of burned areas;•stage II: more or less significant alterations of the vascular wall with necrosis, inflammatory infiltrate and thrombosis; these lesions are located in the peripheral areas of the burns;•stage III: coagulation and necrosis of the entire vascular wall, located in the center of the burns.Rougé and coll  propose,Chaibdraa et al. . In his The brain CT scan showed subarachnoid bleeding .Fig. 6RiThe conduct was stopping anticoagulants with neurological monitoring. The outcome was favorable with resorption as seen on the control CT scan, with resorption of the hematoma and intraventricular hemorrhage .The literature mentions cerebral hemorrhage especially in lightning strikes, but no such observation is reported in industrial current electrification, let alone extradural hematoma.In the end, except in the presence of associated traumatic lesions, there does not seem to be a similar case described in the literature . Electri4Cerebral bleeding due to electric shock are a rare entity described in the literature, their pathophysiological mechanism is still poorly understood, it is difficult to determine that these lesions are due to an accident of electric shock in real life or due to a head trauma associated. A rigorous protection must be made as the helmet and the gloves to avoid this kind of work accident.Written informed consent for publication of their clinical details and/or clinical images was obtained from the patient.Ethical approval has been exempted by our institution.None.Marouane MAKHCHOUNE: Corresponding author and writing the paper.Othman BENHAYOUN: writing the paper.Abdelkouddous LAAIDI: writing the paper.Mohamed yassine HAOUAS: Correcting the paper.Abdessamad NAJA: Correcting the paper.Abdelhakim LAKHDAR: Correcting the paper.None.MAKHCHOUNE MAROUANE.Provenance and peer review Not commissioned, externally peer-reviewed.The authors declare having no conflicts of interest for this article.


This study presented the rare case of a seven-year-old patient with atypical presentation and neuroradiological features of giant ecchordosis physaliphora. The patient underwent cross-sectional imaging due to persistent headache without neurological or visual symptoms. CT scan imaging of the head revealed a hypodense tumor in the prepontine cistern. This lesion caused smooth scalloping of the dorsal clivus without aggressive erosion or calcification, and an osseous stalk was also identified between the lesion and the dorsal wall of the clivus. Sagittal T1 weighted image (T1WI) MRI showed a bilobed, solid and cystic, well-defined lesion, measuring 3.5 cm in terms of craniocaudal diameter, found alongside the midline within the prepontine cistern. After the evaluation of radiological images, the patient was then subjected to endoscopic transnasal complete tumor excision. Histological examination revealed sheets and lobules of clear cells with cytoplasmic globules "physaliphorous cells", and myxoid stroma. There was nuclear pleomorphism associated with focal areas of necrosis. After full recovery and discharge, the patient was followed up for the first year with four-month interval brain MRI scans showing no evidence of residual tumors. On the 12 months follow-up scan, the MRI scan revealed a 1.5 x 0.7 cm recurrent mass in the retroclival right paramidline prepontine cistern, which was most notably seen on the diffusion-weighted images. Series of proton beam therapy with annual MRI scans demonstrated regression of the tumor, eventually allowing the patient to live free of neurological symptoms up to this day. Results suggest that the utilization of radiological imaging such as CT and MRI scans was successful in identifying the ecchordosis physaliphora and differentiating it from chordomas. It can also be inferred that atypical radiological and histopathological findings of ecchordosis physaliphora lesions might suggest that they are prone to recurrence, which is an atypical feature for such entities. Further studies are recommended to explore and better understand these uncommon observations in patients with ecchordosis physaliphora. In 1856, German pathologist Hubert von Luschka first described the presence of pathologic ectopic notochordal tissue at the dorsal clivus  for all Differentiation between ecchordosis physaliphora and its pathologic counterpart chordoma can be achieved based on radiological findings. Ecchordosis physaliphora includes fluid attenuation on CT scan and lack of contrast enhancement or aggressive osseous erosion. Chordomas, on the other hand, typically present as an extra-axial soft tissue hyperdense lesion having osseous erosion, compression on the brain stem (described as a thumb sign), heterogeneous T2 weighted image (T2WI) signal intensity, and varying degrees of contrast enhancement. Intrinsic tumor calcification might be present .This case report presented the atypical presentation and neuroradiological features of a giant ecchordosis physaliphora in a seven-year-old patient.A seven-year-old male patient was subjected to cross-sectional imaging due to persistent headache without neurological or visual symptoms on April 21st, 2016. The patient was medically free until the onset of symptoms and did not have a family history of brain tumors. CT scan imaging revealed a hypodense brain tumor in the prepontine cistern causing smooth scalloping of the dorsal clivus without aggressive erosion or calcification Figures . An osseBrain MRI sagittal T1 weighted image (T1WI) revealed a bilobed, solid and cystic, well-defined lesion, measuring 3.5 cm in terms of craniocaudal diameter, found alongside the midline within the prepontine cistern. This lesion mildly compresses the right side of the pons and displaces the basilar artery. Also, the lesion had an oblong shape and was centered over the posterior border of the clivus intradurally. Analysis of the radiological images revealed the superior part of the cystic component on T1WI to exhibit isointense signal Figure . The isoThe illustration of diffusion-weighted imaging (DWI) and apparent diffusion coefficient (ADC) imaging scans revealed restricted diffusion within the solid part of the lesion Figures -B. HowevAfter the evaluation of radiological images, the patient was then subjected to endoscopic transnasal complete tumor excision. Histological examination revealed sheets and lobules of clear cells with cytoplasmic globules "physaliphorous cells", and myxoid stroma. There was nuclear pleomorphism associated with focal areas of necrosis. Immunohistochemistry staining was positive for AE1/AE3, epithelial membrane antigen (EMA), vimentin, and focally for S100. Ki-67 shows <3% positive staining of the cells in focal areas. Cytokeratin 7 (CK7), cytokeratin 20 (CK20), and CDX-2 staining were negative. The final histopathology report provided the differential diagnosis of notochordal origin tumor and thereby advised correlation with the clinical and radiological results.The postoperative course was uneventful. After recovery and discharge, the patient was regularly followed up for the first year by clinic visits, and four-month interval brain MRI scans, where he remained free from headache or neurological symptoms, and the MRI scan did not show evidence of residual or recurrent tumors Figure .However, on the 12-month follow-up scan, the MRI scan revealed a 1.5 x 0.7 cm recurrent mass in the retroclival right paramidline prepontine cistern, which was most notably seen on the diffusion-weighted images Figure  and sagiConsequently, the primary physician and radiation oncology team, along with the family's approval, decided to begin treatment utilizing a sum of 73.8 Gy (RBE) 41 fractions (1.8 Gy [RBE]) of proton beam irradiation, with planar kV images to ascertain positioning and regular interval contrast MRI during treatment. The clinical target volume was at the skull base region of the recurrent tumor, and a 2 mm planning target volume was included at the margin. Proton therapy was well tolerated without any signs of treatment-related side effects. The first follow-up MRI was obtained three months after completion of therapy and did not show a significant change in the tumor. This was also followed up by a six-month MRI, and later a series of annual MRI scans, auditory, ophthalmological and hormonal tests. Radiological imaging revealed favorable results by the regression of the tumor size. Six years after the initial presentation, the patient is headache-free without neurological symptoms and doing well to this day.The tumor, ecchordosis physaliphora (EP), is an uncommon congenital benign hamartomatous-type which is detected for about 2% of post-mortem examinations ,5. Descr3 in volume or 3 cm in diameter are already referred to as giant EP according to the literature [In terms of classification by size, lesions larger than 6 cmterature . There aterature -13, haviWith regards to diagnosis, EP cannot be differentiated based on histopathological evaluation. Its confirmation is done based on the imaging characteristics ; howeverIn differentiating between EP and chordomas, the observation that they are indistinguishable based on histology and immunohistochemistry supports that both have a common notochordal origin ,7,17,18.This study presented a rare case of a giant ecchordosis physaliphora in a young patient with a unique characteristic of cystic degeneration. The utilization of radiological imaging such as CT and MRI scans was successful in identifying the ecchordosis physaliphora and differentiating it from chordomas. Series of proton beam therapy with annual MRI scans favored the regression of the tumor size, eventually allowing the patient to live free from neurological symptoms up to this day. In addition, results suggest that atypical ecchordosis physaliphora lesions might be prone to recurrence, which is an unusual feature for such entities. Further studies are recommended to explore and better understand these uncommon observations in patients with ecchordosis physaliphora.


Oncocytoma of the lacrimal sac is an extremely rare tumor. In this report, we present the case of an 82-year-old woman who presented with swelling in the region of the lacrimal sac. Systemic examination and ophthalmic examination of both eyes were unremarkable. Computed tomography of the brain and orbits revealed a mass lesion involving the right lacrimal sac with expansion of the related nasolacrimal duct. Neither bone destruction nor tissue invasion was observed. Right external dacryocystectomy and debulking of the tumor were performed. Histopathological examination of the surgical specimen showed oncocytic cells arranged in an adenomatous fashion, and a diagnosis of benign oncocytoma was made. Three years later, the same patient presented with a similar complaint that was pathologically proven to be a recurrent benign oncocytoma of the lacrimal sac. Oncocytic tumors originate from epithelial cells and form secondary to cell aging or functional exhaustion. Oncocytic cells exhibit numerous mitochondria with characteristic histological features of a swollen granular eosinophilic cytoplasm. These cells have a preserved ability to multiply and form daughter cells that maintain their oncocytic features , and oncIn this report, we describe the case of an 82-year-old woman who underwent right external dacryocystectomy for oncocytoma of the lacrimal sac a few years earlier and presented again with a recurrent lesion. Thorough clinical, radiological, and pathological examinations were performed, and the diagnosis of recurrent benign oncocytoma was made.An 82-year-old woman presented with swelling over the region of the right lacrimal sac for one year. Her medical history was significant only for diabetes mellitus type 2. Upon examination, the lesion was soft and bluish, with no tenderness or other signs of inflammation in the region. The fluorescein dye disappearance test was negative in both eyes. Irrigation showed patent canaliculi and nasolacrimal ducts. The patient underwent computed tomography (CT) of the brain and orbits, which showed evidence of a well-defined mass lesion on the right nasolacrimal sac with a density approaching 78 HU. The mass was approximately 2.7 × 2.0 cm and mildly extended and expanded into the related part of the right nasolacrimal sac with a widening of the related part of the right nasolacrimal duct without any evidence of bone destruction or tissue invasion Figures . It had The patient underwent right external dacryocystectomy and debulking of the tumor as it extended beyond the right lacrimal sac; therefore, an attempt to resect all accessible parts of the tumor was performed and sent for pathologic assessment. Postoperative recovery was uneventful. Histopathological examination of the surgical specimen revealed epithelial cells arranged in an adenomatous fashion and in cords . The epiThree years later, our patient presented to the clinic with a complaint of swelling over the right lacrimal sac region that had been growing over the course of eight months . CT of tOncocytic lesions can be hyperplasia, metaplasia, or neoplasia. This article reports the case of a rare benign neoplastic lesion of the lacrimal sac. The clinical features of lacrimal sac oncocytoma range from being asymptomatic to epiphora and chronic dacryocystitis –4. It haOncocytoma of the ocular adnexa more commonly involves the caruncle. A literature review by Say et al. showed that 59% of 212 cases of oncocytic lesions were in the caruncle, 19% were in the lacrimal sac, and only 5% were in the lacrimal gland. The remaining lesions were in the eyelids and conjunctiva. They also have observed that oncocytoma mostly presents in patients older than 40 years and has a duration of symptoms of more than 1 year . AnotherIn general, MRI provides information on anatomical tumor extent, margins, angulation, and configuration of the lacrimal gland fossa mass, while CT is considered the imaging modality of choice to assess bony changes and calcification . Benign The diagnosis is typically based on microscopic evidence of cellular changes that predominantly exhibit oncocytic features. Oncocytosis is known to occur secondary to the degenerative and aging process of epithelial cells. Several mitochondrial DNA mutations have been found to play a role in the transformation of epithelial cells into oncocytic cells. A nationwide study by Østergaard et al. provided the clinicopathological characteristics of oncocytic lesions, which supports the fact that oncocytic lesions have similar immunohistochemical findings to their original cells . This caComplete surgical resection is the treatment of choice for oncocytoma with long-term follow-up for local recurrence. To our knowledge, recurrence after complete surgical resection has not yet been reported. In our case, complete surgical excision was not feasible because the tumor was large and extended beyond the lacrimal sac. The patient presented again three years later with a similar picture of a swelling below the medial canthal tendon of the right side that had been growing for eight months. Postcontrast CT scans showed a similar appearance to the previous mass extending to the preseptal area; therefore, excisional biopsy was performed.


To review and analyze the clinical and imaging features of central giant cell granuloma patients and to review the relevant literatures for the diagnosis and clinical manifestation of central giant cell granuloma.Seven cases of central giant cell granuloma were retrospectively selected for the study, all of which were confirmed by pathology and had relevant imaging investigations. All seven cases had undergone CT scan, three cases had undergone MRI scan. Detailed clinical features were compared along with the imaging findings and analysis was done on the basis of their presentation and imaging features.T1- and T2 weighted images. There may be presence of cystic degeneration, hemorrhage or hemosiderin deposits or osteoid formation, which can cause T1 and T2 signal changes. On contrast study, the lesion doesn’t enhance but periphery may enhance mildly.The clinical features, radiologic features were varied according to the site of the lesion. CT features include unevenly dense expansile mass causing bone destruction and cortical thinning. While MRI features with low to iso-intensity in T1 weighted and T2 weighted images and mildly enhance peripherally, Central giant cell granuloma should be considered.Unevenly dense expansile mass with bone destruction and cortical thinning with low to iso-intensity in  Nowadays, the term reparative has been subsequently been discontinued since the lesion are locally invasive and destructive in nature.1,3,4Central giant cell granuloma (CGCG) is rare and locally invasive intraosseous, non-neoplastic lesion. It consists of cellular fibrous tissue that contains multiple foci of hemorrhage, aggregations of multinucleated giant cells and occasionally trabeculae of woven bone.4,5 Giant cell granuloma is a rare bony lesion in the head and neck region. The mandibular bone is affected in 70% of cases.4 The imaging manifestations in clinical work are often confused with giant cell tumor of bone, aneurysmal bone cyst and brown tumor. In this study, the imaging findings of seven cases of CGCG in different parts are summarized, the aim of which is to improve the understanding of CGCG, to detect and diagnose early, and to choose reasonable treatment methods.Although the etiology and pathogenesis of CGCG is still unknown, it has been believed to be associated with local trauma, repair processes, inflammatory lesion or any development disorders.A total of 19 cases of CGCG were confirmed giant cell granuloma (GCG) by pathology in our  hospital from 2007 to 2019. The institutional review board of our hospital  approved this retrospective study and written informed consent was waived. All of 19 cases were reviewed retrospectively on the basis of their history, clinical features and radiologic descriptions available at our institution and only seven cases were selected for the study, as they had all the relevant imaging and pathological examination findings which included at least one of the radiological investigations with routine Non-enhanced CT scans (NECT), contrast enhanced CT scans (CECT), MRI scans and histopathological investigations.Of these selected seven cases, histories were reviewed in each cases and general data regarding age, gender, presenting symptoms, symptom duration, past history of trauma, lesion size, appearance, radiological findings, laboratory data, treatment modalities and follow-ups were tabulated.T2 weighted image (T2WI), T1 weighted image (T1WI), T2 fluid attenuated inversion recovery image (FLAIR), sagittal T1WI, diffusion-weighted image (DWI) (b = 1000 s/mm2). Gadolinium-diethylenetriamine pentaacetic acid (Gd-DTPA) was used for contrast enhancement.Radiological investigations were reviewed in each case which included CT scan, and MRI with and without contrast, histopathology investigations with hematoxylin and eosin stain. All the patients had undergone CT scan investigation and three patients had MRI scans. CT scan images were obtained by using Siemens Somatom Definition AS 128 sliced CT scanner and non-ionic iodine Iohexol was used as contrast agent for CECT. MRI scans were obtained from GE 3.0 T or Philips 1.5 and 3.0 T MRI scanners. Three patients had undergone MRI scans and image sequences obtained were axial The mean age at diagnosis of CGCG was 22 (standard deviation 15.63) among the seven cases with age ranging from 6 to 46 years. Three cases had GCG lesion in mandible (43%), two cases had lesion in sellar (29%) and one case had lesion in temporal bone (14%) and one case had in maxillary bone (14%). The history of significant trauma to the affected region could not be found in most of the cases (six cases) and only one case had history of trauma to the head could be found.The most common presenting symptoms were swelling and pain. Other symptoms were headache, nausea, vomiting noted in two patients with lesion in sellar region, visual disturbance and protrusion of eye as well as nasal obstruction in one patient with lesion in maxillary sinus. Two patients with mandibular involvement had complains of difficulty in chewing and swallowing.Every case was confirmed central GCG by histopathology. The typical GCG showed clusters of benign giant cells with spindle cell proliferation between the clusters. The cells were arranged loosely without any cell atypia. There was presence of evidences of new bone formation as a result of reactive process. The new bone formed mostly depends on the osteoblastic activity.Every case had undergone at least one of the radiological investigations among CT scan or MRI with or without contrast. All cases showed lesions significant damage to the involved bone. All the cases had well-defined margin and multilocular. The detailed information on each case are summarized in the following All the three cases with lesion in the mandible showed relatively ill-defined, destructive lesion in the mandible causing mandibular ramus destruction along with swollen masseter muscle with low density shadows. Among them, 10-year-old female patient with history of trauma 3 months back and started swelling in face, since then along with difficulty in swallowing, had demonstrated an expansile bony lesion of the left mandible measuring 5 cm x 5 cm x 3 cm originating from the angle of the left mandible . On plaiT1WI ((T2WI (T2 fluid attenuated inversion recovery (FLAIR) image  image  and unifR) image . The masT2WI . It is not a true neoplasm but thought to be as a result of chronic irritation to the area due to local irritation or trauma. Whereas, CGCG are intraosseous and non-proliferative lesions and non-neoplastic lesions. It is less common than PGCG and are commonly manifested in mandible and maxillary bones. Histologically, PGCG and CGCG both are similar. However, they differ in terms of aggressiveness as CGCG are more aggressive and higher recurrence tendency than PGCG.6,8,9GCG are of two forms; peripheral giant cell granuloma (PGCG) and CGCG.1,5 Females are affected slightly more than males, the reason for this is thought to be because of hormonal factors despite the fact that lesions rarely express estrogen receptors.6,10CGCG can display variable clinical presentation, including slow asymptomatic growth without recurrence to fast painful growth with perforation of cortical bone plate and ulceration to the mucosal surface. It can be present in patients from age of 2 years above. But most cases are seen between 20 and 40 years of age.6,11,12CGCG are more commonly located in the anterior portion of mandible and often crosses the midline. But the literatures have reported its occurrence in different locations like hard palate, orbital region, para nasal sinus, nasal cavity and septum, metacarpal bones and phalanges etc, which indicates that it can occur anywhere in the body.12,13 Previously, it was considered to be a hyperplastic reparative reaction to the intraosseous hemorrhage induced by trauma. However, a definite history of trauma may not be reliably elicited. Other theories on pathogenesis of CGCG including infectious and repair process, developmental disturbance, or even inflammatory causes had been proposed, but no single theory has been widely accepted.12 It has also been hypothesized as genetic etiology but lacks the convincing evidence to support the hypothesis.5,6The etiology of CGCG is still not certain and has many theories for its pathogenesis.The most common clinical manifestations of CGCG include pain, swelling and palpable bone lesions and symptoms can vary according to the site of the lesion. In our study, apart from pain and swelling, patients had presented with headache, nausea and vomiting, visual disturbance, protrusion of eyes etc as per the location of the lesion.5,7,11,12 The histologic findings of CGCG, giant cell tumor (GCT) and brown tumor of hyperparathyroidism have virtually identical histologic features and closely resembles granulomas. Immunohistochemical studies have reported CGCG positive for cluster differentiation (CD) 68.5,6 In our study also, four cases of Immunohistochemistry were positive for CD 68.Histological examination of CGCG suggests the lesions are composed of hypercellular fibrous stroma containing numerous multinucleated giant cells within the background of mononuclear stromal cells and spindle shaped fibroblasts along with areas of hemorrhages or foci of cystic degeneration and osteoid production.6,8,12 Chuong et al and Ficarra et al has classified CGCG into aggressive and non-aggressive types on the basis of six criteria like pain, growth rate, swelling, tooth root resorption, cortical perforation and recurrences.14,15 Aggressive lesions exhibit pain and rapid growth and usually more than 5 cm in size with the features of swelling and cortical bone perforation and teeth displacement and root resorption.4,6,9,16 And this type of lesion has high chances of recurrence. Whereas, non-aggressive lesions are low growing and have no or less symptoms and may be without associated features.Radiographically, CGCG appearance ranges from unilocular to multilocular radiolucent well-defined to ill-defined margins. The lesion bony defects size and nature varies according to the aggressiveness of the lesion. Moreover, the lesions may cause damage to adjacent structures like, displacement of teeth, tooth root resorption, cortical bone perforation.16 All lesions appeared to be multilocular and in one case, there was presence of multiple fluid levels within the lesions. The lesions showed heterogeneous soft tissue attenuating masses with mild enhancement on contrast study. MRI have some advantages over CT due to its high soft tissue differentiation and multiplanar depictions. MR images reveal a soft tissue area of low signal intensities on both T1- and T2 weighted images along with variable intensities within lesion if there is presence of fibrosis, osteoid, hemorrhage or hemosiderin deposits. Post-gadolinium MR images of the lesion can show marked enhancement but the degree of enhancement can vary.12 These CT and MR imaging features are not specific but the features can be suggestive of the disease. These imaging features may be indistinguishable from GCT, ABCs and brown tumor of hyperparathyroidism.CT bone algorithm demonstrated that all the patients had the lesions showed lytic bone destruction and with remodeling of adjacent bone. CT scan revealed well-circumscribed expansile mass with the presence of subtle granular bone pattern at the periphery of the expanded bone with some internal septa.5,7,12The differential diagnosis of CGCG includes aneurysmal bone cyst, benign chondroblastoma, brown tumor of hyperparathyroidism, cherubism, fibrous dysplasia, non-osteogenic fibroma, osteosarcoma and true GCT.11Fibrous dysplasia and other odontogenic tumors and non-odontogenic tumors can be easily ruling out on the basis of their clinical and radiological features and histopathology.7,8,11,12Brown cell tumor of hyperthyroidism usually occurs later in the life and is characterized by multiple lesions. Parathyroid hormone, serum and urinary levels of calcium, phosphate and bone or serum alkaline phosphatase are used in the diagnosis of brown cell tumors.6,8,17ABCs are non-neoplastic lesions in the bone containing giant cells. Radiographs show multiple cystic cavities filled with blood within thin walls. MRI reveals a heterogeneous high signal intensity lesion but histologically, they are characterized by thin-walled blood-filled sinuses lined by fibroblasts and giant cells.8,12Chondroblastoma of the temporal bone is a locally aggressive tumor and histologically, it is characterized by presence of hemosiderin pigment, chondroid differentiation, scattered giant cells and calcification and can appear as high-density mass on CT scan.5 It occurs in third to fourth decade of life while symptoms vary according to the site of the lesion. These tumors can go into malignant transformation..5,8,12,18 CGCG and GCT origin are different. CGCG origins from periosteal connective tissue while GCT originates from bone marrow connective tissue. But both the lesions are composed of multinucleated giant cells and small oval or spindle shaped fibroblasts.6,17 CGCG can be differentiated from GCT on the basis of histopathological features as; CGCG have relatively fewer multinucleated giant cells than GCT with increased incidence of osteoid, fresh hemorrhages and hemosiderin deposits. In contrast, the giant cells are more evenly distributed in GCT. Other features of CGGC include increased fibrosis, increased spindle-shaped fibroblasts and absence of necrosis.5,11,12,17 However, considerable overlap of characteristics can occur between these two lesions.While differentiation between GCT and CGCG is often difficult and confusing. GCTs are benign and locally aggressive true neoplasm. They have an incidence of 3–7%. And among them only 2% of GCTs occur in skull.e.g. cryotherapy, osteotomy etc) and resection. All patients in this study had underwent surgical resection and no recurrence cases were observed till now on regular follow-up. Literatures have reported the higher recurrence rate of curettage (33–75%) so, total surgical resection is considered to be the best one for CGCG with lesser recurrence rate (10–20%).7,19,20 Post-surgical morbidity may be high often leading to severe aesthetic and functional problems. Unfortunately, medical management are found to be ineffective in treating CGCG. Different medical approaches including α interferon, calcitonin and intralesional corticosteroid injections have been evolved over last years to avoid mutilating surgery as well as to reduce recurrence after surgical management. Denosumab therapy has been successfully used to treat patients with GCTs of bone and also has shown successful results in treating CGCG.21 However, there is very limited numbers of case studies in this matter, the long-term use of denosumab has shown some promising results. Intranasal calcitonin spray as maintenance therapy has also shown significant result in preventing recurrence after surgical curettage.20,21 Combination of denosumab and intranasal calcitonin may also increase treatment response and reducing in recurrence of CGCG rate, but the sufficient studies are still lacking. Radiotherapy can also be used for CGCG cases where surgery is difficult to perform, but literatures have reported the higher chances of malignant transformation post-radiotherapy.8,9 For the close monitoring of recurrence of CGCG after surgical treatment, CT and MRI should be done on follow-up regularly.Surgical management is the most common treatment modality employed. It can be done by two different procedures: curettage ± adjunctive treatment (T1WI and T2WI and mildly enhanced periphery, CGCG should also be considered.CGCG are non-neoplastic and non-proliferative intraosseous lesions with unclear pathogenesis. It can occur in any part of body and shows symptoms as per the location of the lesion. It can be diagnosed histologically as well as radiographically. But, the disease features are similar to other common diseases like GCT, Brown tumor of hyperthyroidism etc. Radiographically, lesion with unevenly dense expansion along with bone destruction and cortical thinning with low to iso-intensity in


Myxopapillary ependymoma (MPE) is a unique slow-growing benign (WHO grade 1) subtype of spinal cord ependymoma arising predominantly in the filum terminale. Despite its benign nature, it occasionally disseminates through the cerebrospinal fluid and metastasizes to distant sites. Here, we report an extremely rare case of MPE with interval CSF seeding and metachronous metastasis in a 47 –year-old female presented as a gradually increasing low back pain for three months with bilateral radiculopathy down to the knees. Magnetic resonance imaging (MRI) showed an intradural extramedullary spinal mass of iso-intense signal to the cord on T1 weighted-images (WIs), heterogeneous, predominantly hyperintense signal on T2WIs with homogenous enhancement after contrast administration. L2 laminectomy with gross total resection (GTR) was performed, and histopathological results confirmed the diagnosis of MPE. Adjuvant radiotherapy was administered, followed by series of MRI scans. 28 months after GTR, Lumbar MRI showed multiple tiny enhancing nodules in the cauda equina. 44 months follow-up whole spine MRI revealed multiple intradural extramedullary nodules throughout the entire spine. The largest one measures about 1.5cm opposite to T3 –T4 intervertebral disc space. The patient underwent T3 and T4 laminectomy and GTR under general anesthesia using microsurgical techniques, and the histopathological result came with the diagnosis of MPE. Myxopapillary ependymoma (MPE) is a distinct slow-growing benign (WHO grade 1) subtype of spinal cord ependymoma that likely arises from the ependymal cells in the filum terminale. It is the most common primary neoplasm of the conus medullaris and cauda equina . MPE repA 47 –year-old female presented with gradually increasing low back pain for three months with bilateral radiculopathy down to the level of the knees more on the left side. No sphincteric problems, fever, or other complaints. On examination: the patient was conscious, oriented with normal upper and lower limb tone, sensation, and reflexes. Lumbosacral MRI showed an extramedullary, intradural spinal mass at the L2 level that demonstrated an iso-intense signal to the cord on T1 weighted-images (T1WIs), heterogeneous, predominantly high signal on T2-weighted images (T2WIs), and homogenous enhancement after contrast administration. The patient underwent surgical resection with an L2 laminectomy and gross total resection (GTR). Histopathological results were consistent with MPE (WHO grade 1) . AdjuvanPostoperative course: The patient was doing well without any neurologic deficits. Postoperative follow-up MRI confirmed GTR of the neoplasm with no residual tumor.8 months after GTR, follow-up lumbosacral MRI showed no detectable tumor recurrence (currence .Fig. 2T128 months after GTR, follow-up lumbosacral MRI showed tiny enhancing nodules in the cauda equina, but the patient was free of symptoms (symptoms .Fig. 3MR44 months after GTR, the patient presented with 2 months of back pain, progressive right lower limb numbness, and urine incontinence. No fever, lower limb weakness, or other symptoms. On examination, the patient was conscious and oriented but with impaired tandem gait. The lower limb power was 5/5 in all joints except the right hip joint, which was 4/5. Whole spine MRI revealed multiple intradural extramedullary nodules throughout the spine. The largest one measures about 1.5cm at T3 –T4 level (T4 level B. The paAt subsequent follow-up four months after the second surgery, the patient's numbness and urinary symptoms improved. A follow-up MRI showed surgical wound healing ( healing  and mult healing  and 6. A healing .Fig. 5SaMPE is a distinct subtype of spinal cord ependymoma that usually presents as an intradural tumor almost exclusively near the conus medullaris and filum terminale. Although a benign tumor, it can exhibit local recurrence or CSF spread into other regions of the CNS.Cachia et al. Despite the WHO grade 1 classification of MPE, there are few reports of primary CSF dissemination. Khan et al. reported a case of MPE with primary CSF seeding Similar cases of MPE presenting with cauda equina symptoms secondary to CSF seeding presenting several months after primary surgery and adjuvant radiotherapy were reported by Rege et al. Regarding the treatment of MPE, a literature review showed that GTR of the neoplasm is the optimal primary choice An interesting case with striking similarities with ours was reported by Huynh et al. MPE is a benign type of ependymoma that has a potential for local recurrence and CSF seeding even after gross total resection and adjuvant radiotherapy. This behavior is extremely rare but base-line whole spine MRI to detect synchronous CSF seedings appears reasonable, likewise, follow-up imaging, mainly in case of recurrent symptoms.Informed consent was obtained from the patient to publish this case report.


Primary hepatoid adenocarcinoma of the gallbladder is a relatively rare type of extrahepatic adenocarcinoma. The genetic changes involved in this type of adenocarcinoma were unexplained so far. We reported a rare case of primary hepatoid adenocarcinoma of gallbladder with Mab-21 domain containing 2 (MB21D2), polypeptide N-acetylgalactosaminyltransferase 12 (GALNT12), and AT-rich interaction domain 2 (ARID2) mutations, which was confirmed after surgical resection pathologically.A 69-year-old female with distention of hypogastrium and constipation received enema treatment, but ineffectively. No abnormalities were found on relevant physical examination. Then, the CT and MRI demonstrated a 3.3–4-cm soft tissue mass shadow in the neck of the gallbladder. The primary lesions consisted of two components: high-grade intraepithelial neoplasia of glands and hepatoid glands microscopically after laparoscope cholecystectomy. Immunohistochemical staining showed the sameness and difference of the two areas. Furthermore, tumor mutational burden (TMB) shows that the MB21D2, GALNT12, and ARID2 genes were mutated.This is the first report of primary hepatoid adenocarcinoma of the gallbladder with MB21D2, GALNT12, and ARID2 mutations. This will provide a theoretical basis for genetic changes in rare tumors. Hepatoid adenocarcinoma (HAC) is a very rare extrahepatic tumor which originates in the gastrointestinal tract and morphologically and functionally resembles hepatocellular carcinoma (HCC) . HAC couThis is a Chinese case of a 69-year-old female who noticed distention of the hypogastrium accompanied by constipation 20 days ago. No pain, nausea, vomiting, fever, oppression in the chest, jaundice, and bloody stool were found during the period, and she went to the local hospital and received enema treatment. However, distention of the hypogastrium still existed. There were no liver palms, spider-burst, superficial lymph node enlargement, tenderness, rebound tenderness, masses, fluid thrill, shifting dullness, hepatojugular reflux, and pathological reflex on relevant physical examination. The patient denied familial genetic, alcohol abuse, hepatitis, hepatic cirrhosis, psychosocial and exposure to radiation, and toxin history. Furthermore, she was examined in line with the care checklist, as shown in Laboratory tests of the tumor biomarkers , CEA, CA125, CA199, CA153), liver function , and lipid index  of serum were normal.). Unclear cystic duct, dilation of the common bile duct, and bile ducts inside and outside the liver were shown. No dilation of the pancreatic duct or definite abnormality of the liver was detected. Porta hepatis lymph node enlargement was also observed , which demonstrated a 3.3–4-cm soft tissue mass shadow in the neck of the gallbladder which was significantly enhanced on enhanced scan with an unclear boundary to the duodenum and head of pancreas . Hepatocyte paraffin 1 (Hep par-1) showed contrasting results  was used in the case. The MB21D2 gene showed Exon 2E p.Q311E (c.931C > G) mutation, and abundance of mutation was 10.5%. The GALNT12 gene revealed Exon 1 p.L133Q (c.338T > A) mutation, and abundance of mutation was 13.56%. The ARID2 gene indicated Exon 10 p.L381V (c.1141C > G) mutation, and abundance of mutation was 11.47% Table 1.The patient remained under careful observation by radiological and ultrasonic examination, and there was no visceral metastasis in the 5-month follow-up. The patient got an appropriate perspective including the assessment and the episode of care in every month, such as ultrasound examination, rehabilitation training, dietary guidance, regular physical examination, and mental status assessment. The patient healed well, and the distention of hypogastrium disappeared. No adverse or unanticipated events and symptoms in physical examination happened during the period.HAC is defined as a relatively rare tumor consisting of adenocarcinoma admixed with foci of the tumor resembling mature and neoplastic hepatocyte which is proposed as a specific subtype with poor prognosis . In our In this case, CT and MRI revealed that the tumor spanned beyond the neck of the gallbladder. The patient was tested with a normal level of AFP and had no history of alcohol abuse, hepatitis, or hepatic cirrhosis, and thus, prior to liver function, radiologic, and surgery findings, we considered it possible that the origin of this tumor was the gallbladder. Patients with HAC always have markedly elevated circulating levels of AFP , althougImmunohistochemical staining results also supported the HAC. In our case, in the hepatoid gland area, we found negativeness of glypican-3 and Sall-4. A report indicated that glypican-3 and Sall-4 were approximately 87.5% and 93.8% positive in HAC, respectively , which mThe ErbB signaling pathway was related to somatic mutational landscape in gallbladder carcinoma including TP53, KRAS, and ERBB3 . HoweverMB21D2, as a novel cancer gene, which belongs to an intracellular cadherin binder, is found to harbor Q311E recurrent mutation and to be overexpressed in head and neck cancer and involved in cellular processes, including cell survival, proliferation, and migration . MB21D2 GALNT12 is a strong candidate colorectal cancer-susceptibility gene given previous linkage and association studies, inactivating somatic and germline alleles in colorectal cancer patients . GALNT12ARID2 contributes to disruption of the DNA repair process, resulting in susceptibility to carcinogens and potential hypermutation in HCC , especiaIn this case, the challenges of the diagnosis included the rare site of tumor, no obvious symptoms in physical examination, variation of the immune phenotype by immunohistochemical staining, etc. We needed to combine sufficient clinical information, imaging, histological morphology, and multiple immunohistochemical indicators to make the final diagnosis.It is worth noting that the lymph node around the gallbladder had been invaded, which reminded that distant metastasis may occur again in the patient, so we recommend a close follow-up on her. The strength of this case was the specific and rare genetic changes which were firstly reported; unfortunately, we cannot find specific targeted therapeutics for further treatment, and we did not use first-generation sequencing of MB21D2, GALNT12, and ARID2 genes to verify the TMB outcome, which were the limitations of this case.Besides, the patient remained under careful observation, got an appropriate perspective in every month, and received no adverse and unanticipated events after treatment. She felt satisfied and appreciated for the treatment plan, prognosis, and follow-up observation and, furthermore, would continue to be followed up as prescribed. We will continue to monitor the patient’s situation.In summary, we presented an unusual and rare case of HAC in the gallbladder with MB21D2, GALNT12, and ARID2 mutations, which was first reported. Although HAC has characteristic histological features, awareness is important for its diagnosis and prognosis. Therefore, this case will provide a theoretical basis for genetic changes in rare tumors.The patient provided informed consent for the publication of this report and any accompanying images.Informed consent was obtained in this case, and protocols were approved by the Ethics approval of Chongqing University Cancer Hospital and Liangping People's Hospital.ZL contributed to the acquisition, analysis, and interpretation of patient data and the drafting of the manuscript. QJ contributed to the acquisition of the CT and MRI examination data. XC contributed to the HE, immunohistochemistry, and molecular pathological methods. JX and YX gave the final approval of the report. All authors contributed to the article and approved the submitted version.The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.All claims expressed in this article are solely those of the authors and do not necessarily represent those of their affiliated organizations, or those of the publisher, the editors and the reviewers. Any product that may be evaluated in this article, or claim that may be made by its manufacturer, is not guaranteed or endorsed by the publisher.


Juvenile cystic adenomyosis (JCA) represents a rare form of focal adenomyosis in young women.To determine safety and effectiveness of minimally invasive surgery (MIS for JCA).Three patients aged 16-30 years old presented with chronic pelvic pain [2016 - 2019]. Hormonal treatment failed in two cases. Cystic lesions in the myometrium (n=2), and the broad ligament (n=1) was detected on transvaginal 2D ultrasound (TV 2D US) and/or magnetic resonance imaging (MRI). The cyst was separate from the endometrium in all the cases, within the myometrium in two patients and in the right broad ligament in one case. The cystic lesions were confirmed on laparoscopy; and laparoscopic excision of the cysts with adequate repair of the myometrial beds were performed in all cases with fertility preservation. Robotic assistance was chosen in one case in an attempt to avoid injury of the fallopian tube based on the cyst location during a previous laparoscopy. The endometrial cavity was entered in one case.Absence of intraoperative complications and relief of presenting symptoms postoperatively.Pathology report confirmed the diagnosis of JCA is all cases. There were no intraoperative complications. All three patients reported relief of their symptoms 6 to 8 months after surgery. No recurrence of the JCA was reported using TV 2D US in all cases.MIS could be the treatment of choice for patients with JCA. The technique described in our study is safe, effective, and easy to master in experienced hands. Juvenile cystic adenomyosis (JCA) represents a rare form of focal adenomyosis in young women . The firThree nulligravidae aged 16-30 years old presented with chronic pelvic pain (CPP) and dysmenorrhoea. Two of them had tried hormonal treatment without relief. Other causes of pain were ruled out including infectious, genitourinary and gastrointestinal. Table I illustrates the demographic data and a clinical summary of the three cases. Pre-operative imaging and surgical technique is described in each case.The initial pre-operative computerised tomography (CT) scan of the abdomen and pelvis showed a cystic mass separate from the endometrial cavity measuring 2.2 cm x 2.2 cm. A diagnostic laparoscopy undertaken by the referring physician revealed two small endometriotic implants on the left ovary, as well as an endometrioma of the uterus diagnosed via needle aspiration of fluid containing haemosiderin-laden macrophages and blood, with no evidence of malignancy. A hormonal intrauterine device was placed intraoperatively. About one month following her surgery, her pain recurred. The pain was severe, and was not relieved by narcotic medication necessitating immediate intervention. Repeat imaging with transvaginal (TV) 2D ultrasound (US)  and magnTV 2D and 3D US revealed adenomyosis and a cystic mass which appeared to be continuous with the myometrium. A diagnostic laparoscopy that was performed by the referring physician one year earlier showed a 3 cm mass in the right broad ligament under the proximal part of the isthmic portion of the right fallopian tube. The mass was not removed for fear of damage to the fallopian tube. Medical treatment was attempted using oral contraceptive pills for 6 months, followed by a gonadotrophin releasing hormone (GnRH) agonist for 3 months, and unfortunately both failed. During a robotic-assisted laparoscopy by a reproductive surgeon, the uterus, both ovaries, and both fallopian tubes were found to be normal; and the tubes were patent.  A 2 cm rThe initial TV 2D US showed an intrauterine contraceptive device in place, simple right ovarian cysts and a left complex adnexal mass 32 x 32 mm with a differential diagnosis of endometrioma versus ovarian neoplasm. MRI highlighted a 2 x 1.8 x 3 cm ovoid shaped complex cystic area adjacent to the left ovary. A follow-up TV 2D US 5 months later showed a complex left adnexal mass measuring 32 x 32 mm, and a diagnosis of an endometrioma was suggested. Tumour markers including β-HCG, alpha-fetoprotein, LDH and CA 125 were all normal. A diagnostic laparoscopy was performed by a reproductive surgeon.Intra-operatively, the uterus was retroverted, normal in size and contour. The ovaries were normal and both fallopian tubes normal and patent. A left 3 x 3 cm cornual mass was seen. .The same technique of excision of the cystic mass and myometrial closure was performed as described in case 1.  However,All the patients were discharged on post- operative day 1 and their post-operative period were uneventful. Pathology report confirmed the diagnosis of JCA is all cases. All three patients reported relief of their symptoms after 6 to 8 months after surgery with no evidence of recurrence on TV 2D US .The diagnosis of JCA can be challenging. We based our diagnosis in the three cases on the diagnostic criteria suggested by Takeuchi et al.  who reviIt is important to indicate that a noncommunicating horn of a unicornuate uterus with haematometra can be misdiagnosed as a JCA during radiological studies and the correct diagnosis can only be made at time of diagnostic laparoscopy . TherefoPatients with JCA can be managed either medically or surgically. Although there are a couple of reports of successful medical treatment of JCA using a similar approach to the conservative medical therapy for endometriosis, such as using a 12- month course of GnRH agonist therapy, aromatase inhibitors or oral contraceptives in conjunction with acetaminophen and NSAIDs , the majThere are various approaches when considering surgical management for patients affected by JCA. The benefit of laparoscopic minimally invasive surgery and fertility preservation should be employed when possible depending on the surgeon expertise. The first total laparoscopic resection of a cystic adenomyoma in the literature was reported in a 27-year-old woman after unsuccessful laparotomy . The autAnother option for the laparoscopic approach is utilising the robotic system. The first case of robotic- assisted laparoscopic excision in a case of JCA was reported in 2010 and the authors suggested that the robotic-assisted approach would not only be more efficient but also be safer for patients who would like to preserve their reproductive potential . AlthougThe first case of JCA in the literature, which was successfully excised via laparotomy, was described in 1996 . Few othAlternatively, hysteroscopy can be considered for lesions that are more easily accessible through the endometrial cavity. A cystic mass measuring 8 cm with loculations along the posterior wall of the uterine cavity was successfully managed using the hysteroscopic approach . HoweverCystic adenomyoma presents both diagnostic and surgical challenges to the gynaecologist and the approach to therapy depends largely on the patient’s desire for future fertility. Our data and a review of the current literature suggest that definitive surgical treatment, preferably via laparoscopy, should be the initial treatment option. Laparoscopic minimally invasive approach appears to be safe and effective in experienced hands.


Primary pulmonary meningiomas (PPMs) are rare mesodermal tumors that arise in the lung and are most often incidentally identified as single pulmonary nodules. Most cases of PPM are benign, and surgical resection remains the primary curative treatment. We describe the case of a 65-year-old asymptomatic female who presented with an incidentally identified 2.5 x 1.7-cm lobulated, non-calcified mass in the right lower lobe of the lung, which was diagnosed as PPM that had low fluorodeoxyglucose (FDG)-avidity and associated sub-centimeter nodules present in the same lobe. The patient was closely monitored and the nodules showed essentially no interval enlargement over several months. Given the disparate locations and small sizes of the nodules, no surgical resection was planned. The patient remained clinically stable, and close medical monitoring was determined to be the best course of action. Our case highlights the viability of medical monitoring as an alternative to surgery in asymptomatic patients with benign PPMs that have associated micronodules. Meningiomas are the most common central nervous system tumors; however, they may also arise in extracranial sites such as the head, skin, and peripheral nerves. In rare instances, primary pulmonary meningiomas (PPMs) have been identified as ectopic meningiomas arising as primary tumors in the lung -2. So faA 65-year-old female with no significant past medical history was concerned about her family history of cardiac disease and requested to undergo imaging for a coronary artery calcification (CAC) score. The patient had a five-pack-year smoking history and had quit 10 years prior; she was otherwise healthy without any symptomatic complaints. Non-contrast CT of the chest did not reveal identifiable cardiovascular disease but did reveal an incidental right lung nodule. Repeat CT scan and PET scan revealed a 2.5 x 1.7-cm lobulated non-calcified mass in the right posterior sulcus with faint FDG-avidity of maximum standardized uptake value (SUVmax) of 2.127 Figures , 4.Lymph nodes did not demonstrate abnormal metabolism. CT-guided lung biopsy of the right lower lobe posterior mass was subsequently performed and revealed a grossly soft, tan specimen with spindle cell lesions, focal whirling pattern, and presence of psammoma bodies Figure . CytologSince the first case report published in 1983 by Kemnitz et al. , 53 knowHistopathologically, PPMs can be categorized as epithelial, fibrous, or, most commonly, transitional, and they are typically characterized by the presence of spindle cells in whirlpool arrangements and psammoma bodies. Cytology shows the expression of vimentin, EMA, and CD-34 positivity and S-100 and keratin negativity . While aWe described a rare case of an incidentaloma diagnosed as PPM with associated micronodules. Our patient had a 2.5-cm mass in the right lower lobe, showing relatively faint FDG-avidity with SUVmax of 2.127, which was a biopsy-proven PPM with additional smaller micronodules in the right upper and lower lobe with insignificant FDG-avidity. There is a high likelihood that these micronodules may also represent PPMs as there was a slight interval enlargement of a 4-mm nodule; furthermore, pulmonary tumors have been documented to have low FDG-avidity and still have a 24% chance of malignancy . AlthougPPMs are relatively rare causes of incidental solitary pulmonary nodules, and are typically well-circumscribed soft tumors with histopathology displaying spindle cells in whirlpool arrangements and psammoma bodies with vimentin, EMA, and CD-34 positivity. We discussed a case of a patient who presented with these typical features of PPM along with associated sub-centimeter micronodules, all of which showed relatively low or faint FDG-avidity; to date, only a few case reports have described PPM with micronodules. Given the location and distance between the primary tumor and smaller nodules, an extensive discussion regarding the management of the nodules was undertaken, which led to the decision to opt for continued medical monitoring over surgical resection, which is the current standard of treatment for most benign PPMs. We highlight the importance of including PPM in the differential diagnoses of solitary pulmonary nodules and micro-solid nodules, along with a consideration for conservative management in asymptomatic patients with benign tumors.


Background: Giant pituitary adenomas are benign intracranial tumours with a diameter ≥4 cm. Even if hormonally non-functional, they may still cause local extension, leading to symptoms that include mostly gland dysfunction, mass effects, and, much less frequently, apoplexy due to haemorrhage or infarction. Neurological presentation of giant pituitary tumour apoplexy is even more rare and has not been systematically reviewed. Case Presentation: An 81-year-old woman was admitted to the Emergency Department because of acute onset headache, bilateral visual deficit, and altered consciousness. Computed tomography showed a giant mass lesion (>5.5 cm diameter) expanding upward to the suprasellar cistern, optic chiasm, and third ventricle, over-running the sphenoid sinus, and with lateral invasion of the cavernous sinus. Laboratory investigations revealed central adrenal and hypothyroidism insufficiency, while magnetic resonance imaging confirmed a voluminous suprasellar tumour (~6 cm diameter), with signs of pituitary tumour apoplexy. Neurological manifestations and gland-related deficits improved after hormonal replacement therapy with a high dose of intravenous hydrocortisone, followed by oral hydrocortisone and levo-thyroxine. The patient declined surgical treatment and follow-up visit. Conclusions: Giant pituitary tumour apoplexy is a rare but potentially life-threatening condition. Prompt diagnosis and multidisciplinary management may allow a remarkable clinical improvement, as seen in this case. Giant pituitary adenomas are a subset of benign tumours that present mainly as non-functioning pituitary adenomas (NFPAs) and account for approximately 6–10% of all pituitary tumours . AlthougThe clinical spectrum of giant pituitary adenomas varies widely, ranging from totally asymptomatic presentation (and consequently often diagnosed late) to severe hypothalamic and/or pituitary dysfunction and neurological symptoms due to the mass effect. Mechanical compression of the remaining anterior pituitary gland or of the pituitary stem can also result in the gradual development of partial or complete hypopituitarism. Apart from the endocrinological effects, the neurological manifestations secondary to compression of adjacent structures include headache, visual field defects, ophthalmoplegias, and much less frequently, pituitary apoplexy (PA) ,4. PA is thus a rare but life-threatening condition caused by acute infarction or haemorrhage of a pituitary adenoma. NFPAs appear to be at higher risk of apoplexy, with an incidence of 0.2–0.6 events/100 persons per year . It is wThe incidence of PA may vary across different types of pituitary adenoma. For instance, Nielsen et al. retrospectively reviewed 192 consecutive patients with a large suprasellar, clinically inactive, operated adenoma  confirmeHere, we describe the case of a giant NFPA complicated by PA. In this context, there are very few literature reports of giant NFPAs presenting solely with PA. A PubMed-based literature review found that only five case reports ,13,14,15An 81-year-old woman was admitted to the Emergency Department (ED) because of acute-onset headache, altered consciousness, and hematuria. At admission, she appeared lethargic and confused, and her arterial blood pressure was 150/90 mmHg. Neurological examination did not show any sign of meningeal irritation or ophthalmological dysfunction, except for a bilateral vision loss. Apart from the above-mentioned manifestations, the clinical examination was otherwise normal. The patient also suffered from bilateral glaucoma, arterial hypertension, and atrial fibrillation, and was treated with antihypertensive (ramipril) and anticoagulant (acenocoumarol) drugs, respectively. Family and psychosocial history were negative. Routine laboratory test results, including electrolytes and glucose, were normal except for an INR of 8.51, which was compatible with an uncontrolled anticoagulation therapy. An urgent computed tomography (CT) scan of the brain showed a giant mass lesion (>5.5 cm diameter) expanding upward to the suprasellar cistern and the third ventricle, over-running the sphenoid sinus, and with lateral invasion of the cavernous sinus . MagnetiThe conclusion from urgent endocrine evaluation was adrenal and thyroid insufficiency, as shown by a cortisol level of 137 mmol/l , ACTH 3 pg/mL , GH < 0.1 ng/mL , TSH 0.09 µU/mL , fT3 0.56 pmol/l , and fT4 6.47 pmol/l . The level of prolactin was normal at 5 ng/mL . A suspicion of prolactinoma due to the so-called “hook effect” was ruled out by carrying out a dilution test with a serum prolactin sample. Prompt administration of hormonal replacement therapy, comprising a high dose of intravenous (IV) hydrocortisone (administered before the tests results were available) followed by oral hydrocortisone and levo-thyroxine, resulted in remarkable improvement of the neurological and endocrinological manifestations. The patient became fully alert and her confused state and headache both resolved. Because the patient declined surgical treatment, conservative management based on medical therapy and clinical observation was adopted. A short-term clinical, laboratory, and instrumental follow-up was scheduled, but she did not attend the visit.We report here the case of a patient with a rare giant pituitary adenoma (~6 cm diameter) who experienced the even more rare complication of a PA-associated neurological presentation, likely due to an uncontrolled anticoagulation therapy-related haemorrhage. NFPAs are pituitary adenomas without hormonal hypersecretion that may be micro-, macro- or giant adenomas. However, giant pituitary adenomas that present exclusively with PA are considered very rare . Among tAlthough PA mostly occurs spontaneously, precipitating factors can be identified in 10–40% of cases . These iIt should be highlighted that PA as an exclusively presenting symptom is very uncommon in the literature published to date  and endocrinological manifestations . This approach covers not only the increased risk of hypoadrenalism, but also exerts anti-inflammatory and anti-oedematous effects. Since corticotropic deficiency is present in the vast majority of patients at the onset of PA and may be life threatening even if treated surgically or conservatively, IV corticosteroids should be given as soon as the diagnosis is confirmed ,34,35,36Regarding the replacement therapy with GH, there is hardly any information on the treatment with recombinant human growth hormone (rhGH) of old patients with GH deficiency, as was the woman reported here. As previously systematically reviewed , two stuSurgical treatment of pituitary adenomas usually includes both microscopic and endoscopic trans-sphenoid approaches. However, because of their extension and involvement of neurovascular structures, these tumours often pose significant surgical challenges and are associated with increased treatment morbidity and mortality, increased recurrence rates, and overall poor outcome and long-term prognosis ,38. CareFinally, the course of PA is highly variable. When the diagnosis is delayed, and therefore also decompression and corticosteroid treatment, death may occur as a result of adrenal failure or neurological complications. As such, PA should be considered as a medical emergency and any patient with a suspicion of hypoadrenalism needs to start treatment with hydrocortisone . Prompt In this context, the literature review points towards a favourable long-term follow-up in the two cases reported by Perotti et al.  and FanoGiant tumour PA is a rare condition that can present with neurological signs of mass effects and corticotropic deficiency. The case reported here can be viewed as a model for a rare but life-threatening disorder that requires early diagnosis and prompt management, together with close multidisciplinary collaboration.


Teratoma is a rare germ cell testicular cancer composed of cells that are not normally present in the site where it originates. These tumors are rarely described in horses, where they may develop due to cryptorchidism. Teratomas consist of cells originating from different germinal layers, arising from germinal multi-potential cells with differentiation defects, and can simultaneously contain several tissues from two or more embryonic layers. Testicular teratomas are described in horses, cats, dogs, wild boars, bulls, and humans. In the rare descriptions found in literature of testicular teratoma in stallions, they occur frequently in cryptorchid testicles, as a consequence of congenital neoplasm. To our knowledge there is no other report of a dentigerous equine teratoma in a stallion. We describe here a successful laparoscopic removal of a testicular teratoma and its clinic-pathological features. The word teratoma was coined by Rudolph Virchow in 1863 from the Greek words “teras and onkoma” that respectively mean “monster and tumor [swelling]”. Teratoma is a rare embryonic tumor that can be both benign or malignant, most often diagnosed as benign. Teratoma is composed of mature somatic tissue that is not well organized and composed of neoplastic germ cells ,5,6,7,8.To date, there is no effective therapy for this tumor, other than surgical removal. In this report we describe the clinical and histopathological characteristics of a rare case of unilateral abdominal testicular dentigerous teratoma in a horse.TM, Medtronic-Minneapolis, MN, USA) was introduced at portal one into the abdomen with a 10-mm laparoscope 0° inside to provide a safe access. The abdomen was then distended using an automatic high-flow CO2 insufflator up to an intra-abdominal pressure of 8–10 mmHg. The accessory trocars were inserted at portals two and three under laparoscopic supervision. The laparoscope was then placed in the third portal and directed caudally toward the vaginal ring. Only a portion of the mesorchium with the spermatic cord and the ducts deferens was visualized, since the identification of the retained testis was not possible due to the interposition of the large colon. The mesorchium was desensitized with 20 mL of 2% mepivicaine via a laparoscopic needle inserted at the first portal, before starting its manipulation. Ten-millimeter Teeth Claw Jaw Grasping Forceps were inserted through portal two, and placed at the more distal portion of the spermatic cord, which was held under slight tension. A 10-mm LigasureTM was inserted through portal 1 and was applied to seal and transect the spermatic cord and ducts deferens just dorsal to the grasping forceps. The mesorchium, freed of its dorsal attachment, was pulled up, revealing the presence of a large mass, compatible with the right retained testis. The LigasureTM was removed and another of the 10-mm Teeth Claw Jaw Grasping Forceps was placed at portal one; this grasped the spermatic cord as close as possible to the testis, which was then removed from the abdomen by enlarging the incision up to 10 cm. The abdominal cavity was checked for any abnormal bleeding, then it was deflated and the trocars were removed. Portal one was closed in two layers: the external abdominal oblique muscle was closed with size 0 polydioxanone suture and the skin with size 0 polypropylene. Only the skin of portals two and three were closed with size 0 polypropylene in a simple interrupted pattern. The retained testis was submitted for histopathology examination, while the other was left in the scrotal cavity by the will of the owner. Post-operatively, the horse received sodium penicillin  and flunixin meglumine (1.1 mg/kg IV every 24 h) for 5 days as required by our post-operative prevention and prophylaxis protocols, to avoid complications of a mainly infectious nature. The horse was confined then to a stable for one week with daily hand walking. He was then discharged and normal activity resumed.A five-year-old, 450 kg, thoroughbred stallion was admitted for standing laparoscopy for cryptorchidectomy. The left testis was in the scrotum but the right testis was not palpable. The horse was for 48 h and before surgery, sodium penicillin  and flunixin meglumine (1.1 mg/kg IV) were administered as per our pre-operative protocols. The horse was restrained and the right flank was clipped and aseptically prepared for surgery. Sedation was achieved with a continuous rate of infusion of detomidine hydrochloride (8.4 μg/kg IV) and butorphanol tartrate (10 μg/kg IV). The first portal was positioned midway between the tuber coxae and the 18th rib, at the dorsal border of the internal abdominal oblique muscle. The second portal was placed 5 cm ventral and 3 cm caudal to the first one, and the third portal was located in the 17th intercostal space at the level of the ventral aspect of the tuber coxae. A 15-mm skin incision was made at each site for laparoscope and instrument portals, following the infiltration of 20 mL of 2% mepivacaine subcutaneously. A 12-mm trocar-cannula unit . After a necessary cross cut made with a band saw, the cut surface allowed us to detect the presence of four morphologically distinct elements, whitish in color, hard, and apparently normal shaped, referable to well differentiated dental structures, immersed in a hard dentinal-osseous amorphous matrix, which occupied most of the entire cut surface, replacing what must have been the normal structural tissue of the testis .Several tissue samples were collected and fixed in 10% buffered formalin, decalcified with aqueous solution of formic and hydrochloric acid for more than 20 days; subsequently, they were embedded in paraffin, cut into sections of 4–5 µm, and then stained with Hematoxylin-Eosin.In general, the histopathological investigations revealed a neoplastic lesion composed mainly of different types of mature tissues by many germinal layers. In this case bone, enamel, dentin and mineralized areas represented the predominant part of the tumor which were also associated with separate and more marginal foci of mature fibrous tissue and tubular-like and glandular-like structures lined with mucosa, arranged in a markedly disorganized way, unlike the teeth which showed a well-ordered structural organization especially in the enamel and dentin components. In this case, no malignancy was detected at histopathological investigations .Reproductive disorders are common in horses, and cryptorchidism is one of the most common conditions where surgical approaches to remove the testes are the most effective and low risk treatment.Teratomas are tumors consisting of mature and immature tissues derived from pluripotent cells of all three germ layers, and which arise from multipotential germ cells that have undergone partial differentiation. In teratomas, ectodermal , mesodermal  and endodermal  tissues can coexist.Teratoma occurs mostly in cryptorchid testicles in horses, where it is reported in an age range of under one year to a maximum of five years, considering it is most likely congenital ,16,17,18Based on our information, this case-report represents the first case described in literature of a dentigerous equine teratoma in this species. Our bibliographic research found only very few cases of teratomas with ectopic teeth described in the human species, mainly localized in the orbital region or in the lung, and never at testicular level and with related primitive onset. Laparoscopy is the best surgical technique to approach this type of condition because it is minimally invasive and offers good visibility with very few and infrequent complications as described by D. Hendrickson .To our knowledge the surgery was well tolerated by the horse , which i


Arachnoid cyst (AC) is a rare defect of the central nervous system that accounts for 1% of all intracranial lesions, of which only 1% of reported cases are located in the third ventricle. Endocrine manifestations associated with AC include precocious puberty, growth hormone deficiency, and hypothalamic dysfunction. We report a child who presented with a visual field defect, hyponatremia, and precocious puberty related to a third ventricle AC. Hyponatremia as a complication of AC is rare. A literature review revealed two case reports of Syndrome of inappropriate antidiuretic hormone secretion (SIADH) associated with suprasellar AC. The pathophysiology of SIADH in AC is not well understood. Hyponatremia may worsen following endoscopic fenestration of the AC secondary to changes in intracranial pressure. In conclusion, hyponatremia with AC should be recognized during the preoperative and postoperative periods and may require treatment with hypertonic saline in addition to fluid restriction. Arachnoid cysts (AC) are non-cancerous cysts that contain cerebrospinal fluid (CSF) . It is aMost of these lesions are found incidentally while undergoing neuroimaging for other unrelated problems. Moreover, the majority of AC patients are asymptomatic while some may present with headaches, increased head circumference, and developmental delay in pediatric patients. Rarely, due to raised intracranial pressure, the patients may develop symptoms of weakness, seizures, and endocrinopathies, depending on the location of the AC . EndocriA 9-year-old boy presented with decreased vision in the left eye for eight months. He denied any headaches, nausea, or vomiting. His past medical history was unremarkable. On physical examination, his vital signs were stable; height and BMI were at the 90th percentile and weight was at the 94th percentile. He was alert, conscious, and oriented to time, place, and person. Visual exam through Snellen’s chart indicated a loss of vision and impaired visual field in the left eye. He had normal sensory, motor, and reflexes exam of the upper and lower extremities. Pubic and axillary hairs were at the Tanner I stage, testicles were 3 cm bilaterally (Tanner II), and penile length was 7.5 cm.Laboratory tests Table  revealedA brain MRI was ordered for further evaluation of central precocious puberty and hyponatremia. The initial brain MRI with contrast demonstrated a 5.6x4.1x3.2 cm third ventricle AC, severe obstructive hydrocephalus, enlarged sella with a mass effect on the pituitary gland, and deviation of the pituitary stalk along with papilledema . Prior to surgery, his hyponatremia worsened Na 129 mMol/L) and the patient had a significant headache. Treatment with 3% hypertonic saline was given for symptomatic hyponatremia and to prevent further worsening of hyponatremia during surgery. Postoperatively, hyponatremia improved (Na 134 mMol/L) and he was kept on 1200 ml per 24-hour fluid restriction. Three days after discharge, he was readmitted with a history of altered mental status and worsening hyponatremia (Na 128 mMol/L) requiring treatment with 3% hypertonic saline. A low dose ACTH-stimulation test (1 mcg) was performed to rule out adrenal deficiency and revealed normal cortisol response (19.4 mcg/dL). His sodium improved gradually and reached 132 mMol/L (Table 9 mMol/L Suprasellar arachnoid cysts (SACs) are rare; they represent 5-12% of intracranial arachnoid cysts . The actSIADH with SACs is not commonly reported. Our literature review revealed only two case reports of SIADH associated with SAC ,17. The Typically, imaging is used to make the diagnosis of SACs as they are well-defined cysts with a thin wall that displace surrounding structures that follow CSF density on CT and CSF signal intensity on MRI . In our Treatment is usually required for symptomatic patients. If the lesion is asymptomatic, serial imaging and neurologic exams are required to follow up . AccordiHowever, even after successful decompression, hyponatremia may worsen following the endoscopic fenestration of the AC secondary to changes in intracranial pressure . In our SACs associated with SIADH in children are extremely rare and can cause other endocrinologic complications, such as precocious puberty. Additionally, the immediate cyst fenestration could improve the patient's SIADH, as observed in the case above. Moreover, it is important to consider SIADH as a cause of hyponatremia in pediatric patients with SACs.


Intestinal duplications are rare congenital developmental anomalies with an incidence of 0.005-0.025% of births. They are usually identified before 2 years of age and commonly affect the foregut or mid-/hindgut. However, it is very uncommon for these anomalies, to arise in the colon or present during adulthood.Herein, we present a case of a 28-year-old woman with a long-standing history of constipation, tenesmus, and rectal prolapse. Colonoscopy results were normal. An abdominal computed tomography (CT) revealed a diffusely mildly dilated redundant colon, which was prominently stool-filled. The gastrografin enema showed ahaustral mucosal appearance of the sigmoid and descending colon with findings suggestive of tricompartmental pelvic floor prolapse, moderate-size anterior rectocele, and grade 2 sigmoidocele. A laparoscopic exploration was performed, revealing a tubular duplicated colon at the sigmoid level. A sigmoid resection rectopexy was performed. Pathologic examination supported the diagnosis. At 1-month follow-up, the patient was doing well without constipation or rectal prolapse.Tubular colonic duplications are very rare in adults but should be considered in the differential diagnosis of chronic constipation refractory to medical therapy. Due to the non-specific manifestations of this entity, it is rather challenging to make an accurate diagnosis pre-operatively. Surgery remains the mainstay of treatment. Some reports suggest that carcinomas are more prone to develop in colonic/rectal duplications than in other GI tract duplications. They commonly affect children below the age of 2.Imaging studies present no substantial aid in ruling in colonic duplications. In most cases, soft tissue mass extrinsic to the bowel wall is the only finding.Herein, we present a case of a 28-year-old woman with tubular duplication of the sigmoid colon. We describe the clinical presentation, gross examination, and microscopic features associated with this entity and the possible mechanisms underlying the development of this anomaly.A 28-year-old gravida 0 para 0 (G0P0) woman, non-smoker, presented to our institution for chronic constipation. She had a long-standing history of constipation and tenesmus for more than 3 years. She had used multiple laxatives and linaclotide without much benefit. The patient is a physician assistant and mentioned that she eats healthy and drinks plenty of water. She noticed rectal prolapse on a regular basis when she strains, and occasionally, she had to manually evacuate herself for complete defecation. She had no other reported symptoms. Her symptoms got worse a few months prior to her presentation. She denied any family history of colon cancer or inflammatory bowel disease, but her father has irritable bowel syndrome (IBS). The patient has a surgical history of ovarian cyst removal .2. The patient recently had a colonoscopy at another institution, and results demonstrated an entirely examined colon that appeared to be normal on direct and retroflection views. A year prior, a computed tomography (CT) of the abdomen and pelvis revealed a sigmoid colon which appeared prominently stool-filled and an area suggestive of rectal intussusception without wall thickening or peri-colonic inflammatory changes and likely representing the cause of severe constipation and fecal impaction. At that time, she also had a defecography that showed a 3.5 cm anterior rectocele.Vital signs were normal, and routine laboratory tests were within the normal reference range. On physical examination, the abdomen was soft, non-distended, and non-tender. Bowel sounds were normal. Yet, obvious rectal prolapse and anterior rectocele were noted. The patient’s BMI was 23.3 kg/mAt our institution, an X-ray gastrografin enema was performed, showing ahaustral mucosal appearance of the sigmoid and descending colon . MRI defThose findings can be seen with chronic inflammatory conditions or chronic laxative use. The differential diagnosis included IBS, giant colonic pseudo-diverticulum, mesenteric cyst, colonic neoplasia, or colonic duplication. However, IBS was unlikely since the patient’s symptoms were refractory to medical therapy. Due to the severity of her symptoms and failure of conservative management, the patient agreed to undergo a laparoscopic sigmoidectomy with rectopexy for treatment of her rectal prolapse.Diagnostic laparoscopic exploration was performed, revealing a broad-based diverticulum in the sigmoid colon, likely presenting a tubular duplicated colon that shares a common opening with the sigmoid colon .A 17 cm segment of the colon was resected, and a side-to-side functional end-to-end anastomosis was performed. The rectum was straightened and secured to the posterior pelvic wall in order to fix the rectal prolapse. On gross pathologic examination, there was an intestinal outpouching located 4.3 cm from one end of the segment of the resected colon, making a 30 degrees angle with the colon. The intestinal outpouching was 7.3 cm in length and 2.6 cm in average circumference; it had a blind end and appeared grossly unremarkable. On microscopic examination, the intestinal wall of the duplication contained all three layers (true diverticulum) with mucosal lymphoid aggregates that extended to the submucosa . The difIntestinal duplications are rare congenital developmental anomalies.Various theories have been postulated to contribute to the occurrence of intestinal duplications.--The ileum and ileocecal valve regions are the most frequently affected sites. , while the colon is less commonly involved (5-15%).No findings are pathognomonic for intestinal duplications on imaging. Work up can begin with plain chest and abdominal X-rays and can be followed by a CT scan. Ultrasonography, barium enema, and colonoscopy can also aid in the diagnosis. Yet, they might not always yield a definitive diagnosis.Very few cases of tubular sigmoid duplications in adults have been reported in the literature.


Osteochondroma, often referred to as exostosis, is the most common benign bone tumor characterized by a bony protuberance surrounded by a cartilaginous surface. Most osteochondromas are found on the metaphysis of long bones, with the dorsal aspect of the scapula being a rare site of occurrence for an exostosis. Radiographic imaging, preferably through MRI or CT, assists in the identification of benign growth; however, a definitive diagnosis requires a biopsy. Open surgical resection and arthroscopic excision are the definitive treatment modalities of the nidus. Postoperative care requires immobilization of the limb for two months, with at least four months being the appropriate timeline for complete recovery. Osteochondroma is an exostosis defined as a benign bony outgrowth with a cortical layer continuously projecting from the underlying bone, filled with cancellous bone and covered by cartilaginous tissue. Osteochondroma accounts for 35-46% of all benign bone neoplasms. The incidence of osteochondroma under the age of 30 is higher in men compared to women ,2. The oA healthy 23-year-old male presented to the CMH Orthopedic Outpatient Clinic with a complaint of a painless protrusion on the left side of his upper back for the past nine years. Although the protuberance was first noticed by the patient’s mother at age 13, no management was instituted till now. The mass slowly grew till the age of 18, after which its growth was arrested. At presentation, the patient reported a reduced range of motion of the left arm on full abduction. Otherwise, the patient’s history was unremarkable. He reported that his main reasons for visiting were cosmesis and reduced range of motion.On physical examination, a uniform, rounded, protuberant, and sessile nidus was seen and palpated in the medial region of the dorsal aspect of the left scapular blade Figure . AlthougThe patient’s surgery has been scheduled for early 2022 for cosmetic reasons and to prevent future mechanical complaints. He is scheduled to be operated under general anesthesia in the prone position. An open incision will be made along the border of the scapula where the protrusion is most prominent. Muscle traction will be applied, after which extraperiosteal resection of the lesion will be attempted. Along with postoperative care, steroids will be injected into the operative wound to prevent keloid formation. The patient will be followed up for up to one year to monitor for recurrence of the swelling.Osteochondroma is the most commonly occurring benign bone tumor accounting for 8% of all bone tumors. It occurs because of herniated epiphyseal cartilage into the bone through a periosteal defect during the ossification process. Due to this mechanism of growth involving the cartilage, bones that develop through intramembranous ossification have a lower predilection for this tumor. Sessile variants occur more frequently than pedunculated ones. The most common sites of occurrence include long bones (90%) such as the femur, tibia, and humerus . Our patPlain radiographs such as X-rays are sufficient for the diagnosis of osteochondroma in long bones; however, more specialized modalities such as MRI are used to diagnose osteochondroma on flat bones. If not treated appropriately, complications such as pseudo-winging, restricted movement of the shoulder due to fibrosis, snapping, and bursa formation may occur ,13. As oOsteochondroma involving rare locations such as the scapula can be challenging to diagnose for clinicians. Fortunately, our case presented as a bony nidus on the dorsal aspect of the scapula with a recognizable protuberance on gross examination. We recommend including osteochondroma in the differential diagnosis of swelling or pain involving the bony structures of the scapular region.