Gout is a disease characterised by abnormal deposition of monosodium urate crystals, typically affecting the extremities. This report describes a rare case of gout affecting the left temporomandibular joint with erosion of the skull base. A diagnosis of gout was suspected based on CT and MRI and confirmed with CT-guided biopsy. The temporomandibular joint is an uncommon location for a first presentation of gout, with very few cases documented and only three cases of skull base involvement reported in the English literature previously. Given its radiological appearance, it can easily be misdiagnosed as other erosive arthropathies or malignancy. Our paper highlights an unusual location for the first and only manifestation of gout and offers some diagnostic and treatment ideas that may help clinicians to identify and manage this disease. There are only three cases in the English literature where erosion into the skull base is reported and in two of these cases, the patients underwent surgical treatment. The pathophysiology of gout results from hyperuricaemia, either as a result of overproduction, underexcretion or a combination of both. Another closely related condition is pseudogout, which is caused by calcium pyrophosphate dihydrate deposition. Pseudogout of the TMJ has been reported more often in the literature, with seven cases involving erosion into the skull base. Gout of the TMJ presents clinically in a similar way to pseudogout, but the difference is the aetiology and long-term treatment. The treatment for gout of the TMJ involves urate lowering medication as first-line management, and surgery if the patient continues to be symptomatic or has reduced jaw function.Gout is a disease characterised by the saturation of monosodium urate (MSU) crystals in the extracellular fluid, most commonly affecting joints of the lower limb, and rarely the temporomandibular joint (TMJ).Due to the rare nature and the limited literature on gout of the TMJ, clinicians may not consider this diagnosis, resulting in suboptimal or delayed treatment. This case report shows the importance of considering this condition, especially if the skull base is involved to provide appropriate medical and surgical treatment. It presents new information and points of view on a known rare disease, which include diagnosis and treatment of gout of the TMJ.A 77-year-old female was referred to our unit with a 3 year history of constant and progressive left TMJ pain. Her comorbidities include ischaemic heart disease, thyroidectomy for multinodular goitre, asthma, chronic kidney disease, irritable bowel syndrome, atrial fibrillation and anxiety but no history of any inflammatory arthropathies. Her medications include amiodarone, metoprolol, mebeverine, frusemide, thyroxine, venlafaxine, sodium valproate, quetiapine and theophylline. Clinical examination revealed a notable swelling over the left TMJ, that was tender to palpate. She had no neurological deficits and no limitation in mouth opening. There was no palpable cervical lymphadenopathy. She was fully edentulous.−1  and low eGFR of 21 mL/min/1.73 m2 in keeping with her chronic renal impairment. The remainder of the blood examination was unremarkable. A serum urate was not performed as gout was not suspected at that stage.Routine blood examination revealed a CRP of 77.2 mg lCT and MRI were performed without contrast  and revealed a destructive arthropathy of the left TMJ with relatively well-defined articular and juxtaarticular erosions associated with surrounding dense lobulated soft tissue thickening. The erosions caused a through and through defect of the skull base . The adjSodium urate crystals were detected under polarised light microscopy. Histopathology showed mixed acute and chronic inflammation with amorphous basophilic and eosinophilic material. No malignancy was detected. Diagnosis of gout of the left TMJ was made based on microscopy findings.The patient was commenced on a tapering prednisolone regime and planned for urate lowering therapy with allopurinol after 2 weeks and clinical follow-up in 3 months. A total joint replacement was considered concurrently by the treating TMJ surgeon due to the degree of TMJ destruction and skull base erosion. However, the patient had significant comorbidities and surgery was regarded as high risk. Hence, surgery was reserved for persistence or progression of symptoms despite the initial medical therapy as the patient had preserved joint function. The decision on surgery at follow-up will be based on a risk–benefit analysis after re-evaluating the patient. Unfortunately, the patient passed away from an unrelated cardiac event 2 weeks after the diagnosis of gout had been made. The four phases of gout are asymptomatic hyperuricaemia, acute joint arthritis, intercritical gout and chronic tophaceous gout. The classic presentation of gout is an acute monoarthritis affecting the first metatarsophalangeal joint but it can also commonly involve the upper extremities and can progress to be polyarticular. The differential diagnosis of isolated TMJ pain include myofascial pain, internal derangement, trauma, osteoarthritis, rheumatoid arthritis and, less commonly, neoplasms and pseudotumours such as synovial chondromatosis. It is unusual for gout to present in the TMJ, with only 14 cases reported in English literature and 3 involving the skull base.The incidence of gout increases with age and is more common in males and post-menopausal female. In our case, the patient had no previous history of gout and conventional imaging was insufficient to confirm a diagnosis. Malignancy was considered at first. The initial CT and MRI  reports did not include a suspicion for gout and it was only considered after an experienced head and neck radiologist reviewed the images. Conventional CT and MRI are non-invasive techniques that can assess the degree of delineation, arthrosis, erosion and soft-tissue changes, but are not specific in diagnosis unless nodules are calcified and may be mistaken for other diseases like malignancy or pseudogout.The approach to diagnosing gout of the TMJ varies in the literature. Oliveira et al argue that diagnosis can be made with a previous history of gout, presence of hyperuricaemia and imaging, without the need for a biopsy. Some factors affect the feasibility to biopsy, including the general health of the patient and location of the affected joint. Our patient was unsuitable for an open surgical biopsy due to her comorbidities and an unguided biopsy in an outpatient setting would be unsafe due to the deep location of the lesion and potentially nondiagnostic if poorly sampled. Hence, we opted for a CT-guided biopsy of the TMJ. Theoretically, when performing a biopsy of the TMJ, structures at risk of injury include the joint capsule, articular disc and ligaments, however in our patient’s situation, they were already damaged by the pathology and therefore, the benefit of the biopsy far outweighed the potential risk. Generally, inserting the biopsy needle to reach the target lesion carries minimal risk, as the needle tip tends to displace mobile structures like nerves, rather than cutting them. Strategies to optimise pathway to lesion include patient positioning, needle type and performing multimodality imaging. In our case, the patient was positioned in a supine position with the head titled to the opposite side to maximise comfort. A 14-gauge Quick-Core Biopsy Needle was passed through a 13-gauge coaxial guide into the soft tissue thickening. The radiologist ensured the needle tip did not extend beyond the target lesion. This reduced the risk of complications, as when taking biopsies, the specimen notch remains stationary and only the cutting canula moves, closing over the specimen notch and enclosing the sample as it does so  seal the skull base to prevent neurological complications and provide a stable fossa, (ii) replace the condylar head to regain any lost height. Due to the small sample size of gout of the TMJ especially with skull base involvement, we encourage clinicians to report their cases in the literature to build an evidence base about this disease presentation.The therapeutic goals for gout of the TMJ are the same as for gout affecting other joints: pain management with colchicine, steroids and NSAIDs, lowering serum urate levels with allopurinol and regular monitoring for disease progression.In conclusion, this case demonstrates the importance of considering gout in TMJ disease, particularly when image findings demonstrate erosive and destructive changes progressing to the skull base. These cases may be best managed in a centre with experienced head and neck radiologists and TMJ surgeons. CT-guided biopsy, in experienced hands, is quick, minimally invasive and facilitates diagnosis and treatment planning. Surgery should be considered in cases with skull base involvement, severe deformity, and if pain or joint dysfunction persist despite medical management.Gout of the TMJ should be considered in patients presenting with isolated TMJ pain to avoid suboptimal management. It can cause erosion into the skull base, although uncommon.CT-guided biopsy of the TMJ by an experienced head and neck radiologist can be performed instead of a more invasive and time-consuming open surgical biopsy to facilitate diagnosis.Surgery can be considered in patients presenting with skull base involvement, severe deformity, or have failed medical therapy to prevent neurological complications, treat TMJ dysfunction and manage pain.


The primitive neuroectodermal tumor (NEDT) was first described in 1973 by Hart and Earle in order to characterize an undifferentiated neoplasia of the central nervous system, which represents between 90 and 95% of undifferentiated cells and does not fulfill diagnostic criteria for any other type of neoplasia.Our goal with the present paper is to report on a patient who presented peripheral facial paralysis as initial manifestation of the peripheral NEDT involving the temporal bone.R.A.O., a 32 year old female, complaining of 30 days of facial movement impairment, associated to right side otalgia. Otolaryngological exam showed grade IV  peripheral facial paralysis on the right side, and normal otoscopic exam. Temporal bone CT scan showed enlargement of the facial nerve canal within the mastoid . FollowiTemporal bone and facial nerve involvement by neoplasia is not common, it represents about 5% of peripheral facial paralysis causesPeripheral NEDT is a neoplasia of primitive neuroectodermal cells, of rare occurrence. It comes from the peripheral nervous system and bears considerable predilection for limbs and pelvis, regardless of age range.In the literature we found numerous series showing varied primary sites affected by the NEDT.In our bibliographic review (MEDLINE and LILACS) we found no report of temporal bone involvement by NEDT primary or metastatic lesions. Diagnostic confirmation for this tumor is carried out through immunohistochemistry, and it is positive for VimentinePeripheral NEDT is an extremely rare neoplasia, with very few cases described in the literature and without a definitive clinical presentation pattern. This is the first literature report of peripheral facial paralysis as initial NEDT manifestation by involvement of the temporal bone.


An axillary mass was detected in a 6-year-old, neutered, male, domestic short-haired cat during a wellness exam. Gross examination following surgical removal revealed a discrete, deep subcutaneous, discoid mass that was between 0.5- and 0.7-cm-in-diameter and diffusely firm and white. Histologically, the mass was well-demarcated, partially encapsulated, and expanded the panniculus carnosus. It was composed of tightly packed, giant rosettes of radially arranged fusiform cells stacked in one to 10 layers with peripherally palisading nuclei and with centrally oriented, fibrillary, cytoplasmic processes, and collagenous fibers. Laminin immunoreactivity and ultrastructural examination highlighted a continuous basal lamina outside the plasma membrane of each neoplastic cell. Neoplastic cells were immunoreactive for GFAP, S100, periaxin, and Sox-10 and were immunonegative for synaptophysin, smooth muscle actin, and pancytokeratin. Collective findings were consistent with a diagnosis of neuroblastoma-like schwannoma. This is the first veterinary report of this rare variant of benign schwannoma. In cats, cutaneous schwannomas, which are grouped within the entity of benign peripheral nerve sheath tumors (PNST), occur predominantly in the head or neck and, with less frequency, in the limbs or the trunk . Recent A 6-year-old, neutered, male, domestic short-haired cat was presented to the William R. Pritchard Veterinary Medical Teaching Hospital (VMTH), University of California – Davis, California, USA, for a wellness examination, including an evaluation of an axillary mass that was first noted by the owners 2 weeks prior to the visit. Prior medical history included feline herpes virus-1 infection and symblepharon of the left eye as a kitten, and mild overconditioning and mild dental disease as an adult.The mass was palpated as approximately 1-cm-in-diameter, round, firm and partially adhered to the muscle and connective tissue of the left lateral thoracic wall and axilla. Examination of a fine needle aspirate of the mass was tentatively diagnosed as a feline basal cell tumor and the owners elected surgical removal of the mass. It was removed ~1 week later with 1- to 2-cm lateral margins and with one fascial plane deep to the mass. At surgery, the mass was discoid, between 0.5- and 0.7-cm-in-diameter and diffusely firm and white. The fresh specimen was trimmed, and most sections were immediately immersion-fixed in 10% buffered formalin for 24 h and then processed and embedded in paraffin wax. Additional tissue sections were submitted in Karnovsky's fixative to California Animal Health and Food Safety Services Laboratory Systems  and routinely processed for transmission electron microscopic examination. For light microscopy, 5-μm tissue sections were stained with haematoxylin and eosin (H&E), Masson's trichrome stain or processed for immunohistochemistry (IHC) using 1-h incubation periods with one of several primary antibodies . The panHistologically, there was a discrete, multilobular and partially encapsulated mass that focally expanded the panniculus carnosus in the vicinity of hypercellular peripheral nerve fascicles. The stretched capsule surrounding the mass consisted of epineurium and a few residual nerve fibers . The lobA final diagnosis of neuroblastoma-like schwannoma was made based on the cytomorphological features and the strong immunoreactivity to laminin, which characteristically highlighted basal lamina around each neoplastic Schwann cell. Additional immune markers revealed that neoplastic cells were also strongly immunoreactive to GFAP, S-100, periaxin, and Sox-10 but with no immunoreactivity to synaptophysin and SMA . The proRosette formation with strong immunolabeling of neoplastic cells with neuronal-specific immune biomarkers such as synaptophysin, is a characteristic feature of neuroblastomas . This laIn summary, this is the first report of neuroblastoma-like schwannoma in a domestic animal. The histologic hallmark of this variant of schwannomas is the uniform rosette histoarchitecture. Despite they can resemble Homer-Wright rosettes, they are much larger than those seen in neuroblastomas, lack immunoreactivity to synaptophysin and display strong immunoreactivity to Schwann cell markers like laminin, S-100 and Sox-10. Also, their centers appeared to be composed of collagen rather than neurites, as demonstrated by histochemical stains and immunohistochemistry. Histological features that favored a benign diagnosis in this case include encapsulation, lack of mitotic figures and absence of necrosis. At a 3-month follow-up examination, a computerized tomography (CT) scan demonstrated mild contrast enhancement of the left ventrolateral thoracic body wall subcutaneous tissue deep to the previous surgical scar, suggestive of either granulation tissue or recurrent neoplasia. However, further contact for follow-up with the cat has been lost.The raw data supporting the conclusions of this article will be made available by the authors, without undue reservation.VC was the UC Davis anatomic-pathology resident who received and work-out the case. PM-S was a visiting Ph.D. student who work-out the IHC of the case. SS was the UC Davis Neuropathology Fellow who act as the reviewer neuropathologist and lead the diagnostic immunohistochemical approach. RH and AB were the expert opinion senior neuropathologists who help diagnose the case. All authors contributed to the diagnosis of the case and concept of the case report and writing.This study was initiated and completed at the VMTH of UC Davis during VC's last year of residency in anatomic pathology and SS as a Fellow in Neuropathology, UC Davis, Davis. This study was funded by the Center for Companion Animal Health at UC Davis (CCAH Alloc.# 2013-35-R siso). PM-S was supported by an AGAUR Ph.D. visiting grant [2012 BE100584] from the Generalitat de Catalunya, Barcelona, Catalunya, Spain.VC was employed by Charles River Laboratories, Inc. The remaining 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. 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Teaching Point: A chondroid lipoma is a rare benign adipocytic neoplasm, containing a chondromyxoid matrix, that may mimic a myxoid liposarcoma and myxoid chondrosarcoma. Figure 1A–B: sagittal bone and soft tissue window, arrow). There was no invasion of the lesion in the surrounding structures. Subsequent magnetic resonance imaging (MRI) of the left hand was performed for further grading, characterization, and defining the local extent. MRI confirmed a heterogeneous soft tissue mass located in the adductor pollicis muscle. On the palmar side, the lesion was of high signal intensity on T1-weighted images (WI) in keeping with fat . In the deep part, foci of low and high signal were seen on fat-suppressed T2-WI . Suppression of the fat signal was seen at the palmar part of the lesion. The areas of low signal correlate with calcifications on CT, whereas the areas of high signal are suggestive for chondromyxoid matrix. The lesion showed delayed and weak enhancement at the periphery of the mass . The diagnosis of a chondroid lipoma was suggested. Because a malignant lesion could not be excluded, the lesion was resected. Histopathological examination confirmed the diagnosis of a chondroid lipoma.A 79-year-old man presented with a painful swelling in the left hypothenar region, that he noticed for two weeks. Computed tomography (CT) revealed a well-circumscribed mass, located in the hypothenar muscles. The lesion was heterogeneous, containing arciform calcifications, ossifications, and areas of fat and soft tissue density (A chondroid lipoma is a rare benign adipocytic neoplasm, that presents as a well-circumscribed lesion located in the subcutaneous tissue, superficial muscle fascia or skeletal muscle. It is mostly located in the proximal extremities and has a female predilection. Patients mostly present with a painless, slowly growing lump. Cytogenetic aberrations have been reported associated with a translation between chromosome 11 and 16. Macroscopically, it contains a mixture of fat, chondromyxoid matrix, and metaplastic calcifications or/and ossifications.Imaging reflects the macroscopic appearance of the lesion. Arciform calcifications, due to calcified chondroid matrix, are usually seen on radiographs and CT. On MRI, calcifications are of low signal on all pulse sequences, whereas non calcified chondromyxoid matrix is of high signal on T2-WI. Fatty components are isointense to subcutaneous fat . The com


Gastrointestinal stromal tumours (GIST) occur more commonly in the stomach and make up ~80% of the GI mesenchymal neoplasms. They are very rare in young adults and in males. The diagnosis is confirmed histologically and immunohistochemically. Once diagnosed, survival rates are dependent on various factors. The main treatment is resection, but targeted therapy can be used pre or post-operatively. This case is of a 35-year-old female with no significant medical history presenting to her general practitioner with lethargy, malaise and mild weight loss. Initially, she was investigated for a haematological malignancy, but upon further investigations, her computed tomography (CT)  scan showed an abdominal mass, which was resected and found to be a high-grade perforated gastrointestinal stromal tumour in her proximal ileum. This is a good example of an atypical presentation of GIST and emphasizes the importance of thorough workup and prompt surgical intervention in achieving a satisfactory outcome. Gastrointestinal stromal tumours (GIST) occur in the gastrointestinal (GI) tract due to proliferation of the GI mesenchymal cells. This is due to the abnormal form of tyrosine protein kinase (KIT) known as CD117. KIT up regulates stem cell precursors of the interstitial cells of Cajal, resulting in tumour formation. The tumours originate anywhere from the oesophagus to the anus . They maPatients are usually asymptomatic and the GIST is found incidentally or on exploration . The sevGI bleeding can occur in half of the cases and presents with symptoms of anaemia, melaena or haematemesis. GI bleeding and abdominal pain are more commonly associated with gastric GIST, whereas acute abdominal symptoms are associated with jejunal/ileal GISTs . RuptureComputed tomography (CT) is the gold standard modality for determining the size, location and spread. Positron emission tomography can also be useful in the localization of secondary lesions . HoweverThe goal standard treatment is surgical resection. In patients with locally advanced tumours or metastasis, adjuvant targeted therapy using tyrosine kinase inhibitors (Imatinib) can be used .This case report presents an atypical presentation of GIST in a young, previously well female who presented with a general feeling of fatigue, lower abdominal pain with anaemia, thrombocytosis and lymphocytosis. CT of abdomen and pelvis showed a small bowl mass for which she underwent a semiurgent laparotomy . The post-operative histology later confirmed a perforated GIST, which is a rare complication of GIST .A 35-year-old female presented to the hospital with lethargy, exertional dyspnoea, anorexia and weight loss. This was accompanied by vague abdominal symptoms and lower abdominal discomfort. One week prior, she reported of having fever and supra pubic pain, which was treated for a suspected urinary tract infection.Her medical history included cobalamin deficiency and seasonal allergies. Drug history included four monthly B12 injections. She was a non-smoker and her alcohol consumption was reported minimal. She was independent with her daily activities.Her routine blood tests showed that she had a normocytic anaemia (Hb = 70 g/l). Her iron studies were normal, B12 was high and folic acid was normal.9 cells/l), platelets (1183 × 109 cells/l) and C reactive protein of 108 mg/l. She was subsequently referred to the haematology department for a suspected myeloproliferative disorder.She had a raised white blood cell count . The scan reported a lower abdominal/pelvic thick-walled fluid and gas-filled structure that measured 5 × 7 × 6 cm lying adjacent to the urinary bladder with a likely fistulous tract to the overlying loops of bowel. A small amount of free fluid and non-specific stranding of fat was seen in the pelvis. No pulmonary or metastatic nodules were seen  and 2.The haematology team subsequently referred her to the general surgeons. Surgical review revealed a pale patient, with sinus tachycardia of 94/min. Remaining observations were unremarkable. Abdominal examination revealed a non-distended, soft, mildly tender lower abdomen with no visceromegaly. Keeping in view of the history, clinical findings, blood picture and the results of the CT CAP, she was teed up for a semiurgent laparotomy. Differential diagnosis included Meckel’s diverticulum, tubo-ovarion pathology and small bowl lymphoma. Her preoperative morbidity and mortality according to National Emergency Laparotomy Audit score was 10 and 1.5%, respectively.Laparotomy revealed localized pelvic sepsis, a large perforated inflammatory mass arising from the proximal ileum firmly adherent to the dome of the urinary bladder and the sigmoid colon. Because of the significant inflammatory fibrosis, with no planes of dissection, the small bowl mass was mobilized off the urinary bladder and the sigmoid colon via blunt dissection. The mass was resected via a small bowl resection and a primary hand-sewn, double-layered anastomosis was fashioned. Thorough lavage of the peritoneal cavity was performed with normal saline. Specimen was sent for histology and immunohistochemistry.The patient made an uneventful post-operative recovery with no residual symptoms. The histology came back reporting a ruptured GIST measuring 60 × 70 × 60 mm in size with a 3a prognostic GIST category according to the AFIP risk stratification scheme .Immunohistochemistry showed diffused CD117 positivity and subtotal immunopositivity of CD34 and SMA .The patient was then subsequently referred to a tertiary centre multi-disciplinary team meeting for further management.GISTs can present in an atypical fashion and can be difficult to diagnose. Clinicians should be aware of the latter. This case report was a good example of an atypical presentation of GIST, which was initially thought to be a haematological malignancy. However, a good outcome can be achieved through thorough investigations and prompt surgical intervention.


Neurilemmomas are tumors of neural origin that comprise Schwann cell proliferation in a characteristic pattern. They are benign in nature. Ancient neurilemmomas are usually longstanding growths that exhibit degenerative features that could be mistaken for malignancy. We report a case of ancient neurilemmoma in a 70-year-old male patient in the retroperitoneal area. Retroperitoneal schwannomas are extremely uncommon along with ancient neurilemmoma features making it worth reporting. Ancient neurilemmomas are tumors of a rare category. These originate from peri-neural Schwann cells. The term “ancient” denotes degenerative features of hemorrhage, cyst formation, hyalinization, and calcification acquired due to advancing age in these tumors . These aClinical findingsA man aged seventy years presented with chief complaints of fever and headache for a few days. He also had a complaint of lower back and bilateral limb pain for four months. The routine physical examination was normal, and the abdominal examination showed no positive findings. The blood examination reports revealed that the patient was found to be positive for malaria  and dengue, which explained the constitutional symptoms. However, chronic bilateral lower limb and lower back pain prompted the further radiological investigation. The patient was started on antimalarials and treatment for dengue fever along with conservative management of pain and radiologic investigation during the first admission showed mild disc bulge along with an incidental mass in the left retroperitoneal region. The patient was kept under observation for the incidental radiologic findings till the management of the diagnosed infection. The patient was discharged after treatment for malaria and dengue and came for a follow-up visit after one month with persistent abdominal pain radiating to the back that was not relieved by pain medication prescribed during the first admission. Hence, radiologic investigation during this visit showed a growing mass, and the patient was admitted for evaluation and surgical removal.Radiologic findingsUltrasonography (USG) of the abdomen was ordered during the first admission to rule out hepatosplenomegaly but showed no positive findings. However, USG findings rather showed an approximately 5.5 × 4.3 cm sized heterogeneous echotexture lesion, predominantly hypoechoic with a few anechoic areas in it with minimal internal vascularity just medial to the lower pole of the left kidney in the retroperitoneal region. An MRI scan was also ordered during the first admission showing marginal peridiscal osteophytes at the lumbar vertebra with subtle Modic type-2 regenerator changes at adjacent cortical plates and diffuse disc bulge with left paracentral disc extrusion at the L5-S1 level, causing compression over the left transverse nerve root along with mild diffuse disc bulge at L2-L3 level without significant neural compression. The scans also showed a well-defined incidental heterogeneous lesion measuring 4.8 × 4.8 cm seen adjacent to the left anterior surface of the left psoas muscle in the left retroperitoneal space. During the follow-up visit after one month for the incidental radiologic findings, evaluation on USG showed an increase in the size of the retroperitoneal lesion to approximately 6.4 × 4.3 cm having mixed echogenic lesion with a few anechoic cystic areas, in its suggestive of necrosis with minimal internal vascularity and foci of calcification noted in the para-aortic region on the left side adjacent to the lower pole of the left kidney  guided percutaneous tru-cut needle biopsy of the mass was taken during the second admission after one month of the initial incidental finding to further assess the pathology and rule out malignancy. The report suggested predominant fibro-collagenous tissue with a few benign spindle cells along with focal areas of chronic inflammatory infiltrate consisting of lymphocytes likely to be a schwannoma Figure .ManagementExploratory laparotomy done during the second admission after four days of positive biopsy findings showed a mass medial to the left kidney adjacent to the L2-L3 vertebrae, which was excised en bloc with no signs of invasion in the surrounding tissues. The excised mass was sent for histopathologic examination, whose results confirmed the biopsy findings Figure .Retroperitoneal schwannomas are such rare tumors that they make up only about 1-5% of all masses in the retroperitoneal region. The tumor localization in the retroperitoneal region accounts for 0.75-2.6% of all schwannomas, while the majority are localized in the extremities, head and neck. These are usually solid, firm, and solitary, with a well-circumscribed smooth surface. They are large and encapsulated tumors originating from the paravertebral region. Macroscopically, schwannomas tend to undergo spontaneous degeneration with hemorrhage more than those in the head and neck and extremities. Prevalence is higher in the older age group, with patients usually not being diagnosed before the age of 40-60, also the male-female ratio of these tumors is almost similar, which is roughly 2:3 in proportion [The tumor is generally encapsulated and, on microscopy, we mainly find two main components: a cellular region arranged in order (Antoni A area) and a loosely arranged myxoid component (Antoni B area). The cellular regions are sclerosed or fibrotic, and over time, hematomas and cysts develop due to degenerative changes. Nuclear palisades are absent, which are normally present in classic schwannoma. Common findings like nuclear atypia and hyperchromasia were present in this case. Malignancy can be ruled out in the absence of mitoses and the presence of a preserved cohesive clustering of spindle-shaped cells. The neural origin of these tumors results in diffuse positivity on immunostaining with S-100 . One of Radiological imaging is important as it helps in guiding treatment plans and also provides information related to tumor size, location, and invasiveness to other structures. Spinal cord tumors or tumors surrounding the sciatic or femoral plexus can cause compression of nerves and give clinical findings similar to herniation of the vertebral disc as seen in this patient. Hence, the radiologic investigation becomes of utmost importance as such tumors are likely to be misdiagnosed as discal herniation, which could delay appropriate management . UltrasoNeurofibroma, liposarcoma, pheochromocytoma, paraganglioma, malignant fibrous histiocytoma, hematoma, and lymphangioma are among the difficult differential diagnoses for retroperitoneal schwannomas . There iAncient neurilemmoma in the retroperitoneal region is an uncommon benign variant of schwannoma with rare malignant development. In our situation, it was diagnosed incidentally on the radiologic investigation done to evaluate symptoms suggestive of lumbar radiculopathy. Usually, schwannomas are asymptomatic slow-growing tumors but considering the location adjacent to the vertebral column in the retroperitoneum, even a slight increase in size, as was in this case, can cause compression symptoms mimicking radiculopathy. However, since typical symptoms of radiculopathy were not characteristically present and the incidental mass in that very region was increasing in size, it was speculated that the growing tumor was contributing to the patient's chronic presenting symptoms and hence its removal became imperative. The tumor was effectively removed by exploratory laparotomy with no complications and a positive outcome with a significant reduction of pain was anticipated.


Thyroglossal duct cysts are a common congenital anomaly in the neck which usually present in adulthood as a midline neck swelling the location of which can vary from lingual, suprahyoid, infrahyoid and suprasternal. Here we have described the case of a thirty-year-old male who presented with a history of recent onset dysphonia and an unnoticed neck swelling. Clinical examination revealed a midline 2 x 2 cm cystic lesion in the neck. On further evaluation using laryngoscopy and computed tomography the patient was found to have a smooth mucosa-covered cyst in the subglottic location. Surgical excision of the cyst was done via laryngofissure and the postoperative course was uneventful. The postoperative biopsy revealed a subglottic thyroglossal cyst. Though there have been reports of intralaryngeal extension of thyroglossal duct cysts, the subglottic location is extremely rare. Through this case report, we would like to highlight the atypical presentation of thyroglossal duct cysts and how an innocuous pathology can turn potentially life-threatening. We would also recommend avoiding a fine needle aspiration cytology in such cases due to the critical location. Thyroglossal duct cyst is a developmental anomaly found in 7% of the population . AlthougA thirty-year-old male presented to the outpatient department with a one-month history of dysphonia, following an episode of upper respiratory tract infection. He was a nonsmoker and had no history of throat pain, cough, difficulty in breathing or swallowing. On clinical examination, there was a 2x2 cm midline neck swelling which was unnoticed by the patient. The swelling was a well-defined cystic swelling which moved with deglutition and with protrusion of the tongue. Laryngoscopy revealed an anterior subglottic submucosal cyst with mobile vocal cords Computed tomography revealed an ovoid well-defined non-enhancing cystic lesion in the anterior midline of the neck of size 2.5x2.8x2.1cm between the thyroid and cricoid cartilage with scalloping of the superior aspect of the cricoid. Posteriorly the lesion projected into the subglottic region and upper trachea causing mild compromise of the airway. Due to the critical location of the cyst a Fine needle aspiration cytology (FNAC) was not performed in this case .He underwent laryngofissure with cyst excision under general anaesthesia. A 2x2 centimetre cystic swelling was delineated by methylene blue dye injection in the midline between the thyroid and cricoid cartilage extending posteriorly to the ala of the thyroid cartilage. The entire cyst was excised in toto without any complications and was sent for histopathologic examination. Gross examination revealed a dye-stained collapsed and cut opened cystic specimen measuring 2x1.5x1cm. Microscopy revealed a cyst wall lined by respiratory epithelial lining with focal squamous metaplasia. Foci of ulceration showing granulation tissue were noted. The wall was fibro collagenous with few blood vessels, mild mixed inflammatory cell infiltrate and occasional glands. There were focal fibrosis and myxoid changes. The histopathologic examination was consistent with a thyroglossal cyst . The patient did not have any postoperative complications and was discharged on oral antibiotics on postoperative day 2. Following surgery patient is asymptomatic and on regular follow-up.During the fourth week of fetal development, the thyroglossal duct develops from the epithelium on the floor of the pharynx at the site of the foramen caecum which lies at the junction of the anterior two-thirds and posterior one-third of the tongue. This duct descends caudally through the neck, in the midline anterior to the hyoid bone and the bilobed distal end differentiates into the thyroid. This duct normally undergoes involution or atrophy by the eighth or tenth week of intrauterine life. The incomplete involution of the thyroglossal duct remnant during embryogenesis leads to the formation of a thyroglossal duct cyst (TDC). It can be formed anywhere along its pathway of descent from the foramen caecum to the suprasternum and the most common presentation is that of an anterior neck swelling that moves with deglutition and with protrusion of the tongue. Although most are situated in the midline, 10 to 24% of the cysts are located laterally, usually to the left (This has been explained by the fact that levator glandulae thyroideae muscle is ordinarily found to the left. A lateral position may be expected especially in the region of the thyroid cartilage because the shape of the thyroid cartilage is like the prow of a ship, on which the cyst cannot remain. The tract may remain silent for several years and may present symptomatically due to inflammation, infection or mucous retention . AccordiThrough this case report we would like to highlight the atypical presentation of thyroglossal duct cysts and besides its malignant potential, how an innocuous pathology can turn potentially life-threatening by causing airway compromise if ignored. Thus thyroglossal duct cyst should be considered as a differential diagnosis of subglottic cysts and FNAC should be deferred in such cases due to the risk of airway obstruction.


Chordoma is a rare, malignant neoplasm thought to develop from the notochord. It most commonly occurs in the base of the cranium or the sacro-coccygeal region but around 15%-20% affect the vertebral body. Extra-lesional resection with or without adjuvant radiotherapy is generally accepted as the mainstay of treatment for this slow-growing tumor. We present a case whereby a patient with an extensive vertebral body lesion causing caudal compression, treated with spinal decompression and posterior stabilization. This case highlights the importance of pre-operative tissue diagnosis, and that, although rare , chordoma should always be considered. We present an extensive case of lumbar chordoma causing tail compression, initially thought to be spinal echinococcosis based on morbidity and radiological studies. Therefore, this patient underwent a 2-stage procedure, first with posterior stabilization of spinal decompression, at which point extralesional resection was more appropriate.A 28-year-old trainer presented with a 6-month history of back pain and increased leg pain and weakness. (Foot numbness? Foot pain?). He reported no history of trauma. His history is unremarkable. Lumbar radiographs showed sclerotic L2 and destructive L1 vertebral bodies associated with osteolytic destruction of the L1-L2-L3 pedicles. There was no evidence of a paravertebral mass .Fig. 1RaMagnetic resonance imaging (MRI) showed extensive heterogeneous bone-based malignancy in the paravertebral space extending from L1 to L3 with hypointense T1 signal and hyperintense T2 signal, without significant enhancement after gadolinium injection. It results in severe canal stenosis with posterior elements extending posteriorly to the left posterior arch of L3 and anteriorly to the psoas and abdomen  and 3.FiSince the patient was a dog handler and based on frequency and radiological studies, the most likely diagnosis was spinal hydatid disease.Due to the large size of the chordoma, a double-approach resection was planned. The first was a posterior approach consisting of a D11-L5 instrument and his L1-L2-L3 left hemilamine resection. An anterior approach was performed 12 weeks after his posterior approach. A multilobed tissue tumor was found at retroperitoneal exposure. Aspiration revealed vesicular structures potentially consistent with echinococcosis. Apsoas resection, L1-L2 hemisection, and partial resection of the L3 superior endplate were performed. The bone defect was reconstructed with a non-vascularized fibular graft from the left leg. The patient survived the procedure without any problems and received albendazole as adjuvant drug therapy. One year later, the patient was asymptomatic, disease-free, with an acceptable level of activity and a stable spine .Fig. 4PrHistopathological inspection of the resected mass revealed multiple varying sized nodules of large, pale, multivacuolated physaliphorous cells showing mild nuclear pleomorphism, with some areas of metaplastic cartilage formation within the tumor. Immunohistochemistry was positive for CK, PS100, vimentine and EMA staining (C68 negative) . HistoloChordoma is a primary bone tumor that arises from remnants of the notochord is characterized by slow growth over years and often spreads to the lungs, bones, soft tissues, liver, skin, or lymph nodes. It is associated with late metastases in 40%-60% ,2.The 5-1.Clinical symptoms are generally delayed. In other words, chordoma is often large at diagnosis and other oncological treatments are inappropriate ,3,6.2.Poor surgical margins are associated with poor survival and a high recurrence rates3.Local tumor progression is a strong prognostic factor that reduces survival ,5.4.Chordoma is resistant to conventional radiotherapy (cured only at toxic doses [60 Gy]) and chemotherapy Symptoms most commonly result from compression of the anterior columns and roots. The most common complaints of patients with lumbar chordoma are pain and paresthesia. Motor changes can occur, but paraplegia is rare However, in most scenarios, marginless resection is difficult due to the surrounding neurovasculature and extensive tumor size ,5. RadiaEn bloc resection can improve survival and reduce the rate of local recurrence, and is often used to treat chordoma . En blocIn our case, the patient underwent a combination of anterior and posterior L1-L2 stage hemirectomy and partial resection of the L3 upper endplate. No studies have compared the effectiveness of the combined posterior and anterior-posterior approaches. Theoretically, a posterior approach alone is inadequate for direct visualization of abdominal structures and increases the likelihood of major vascular injury. The combined approach has the disadvantage of longer operative time for patients, but it can be done alternately or simultaneously. Due to the extensive nature of our patient's chordoma, a stepwise approach was most appropriate. The overall complication rate after total en bloc resection is 36.3% in most studies. Complications include severe neurovascular injury, excessive bleeding, tumor cell contamination, spinal instability, and abdominal herniation (after anterior approach) .ResectioLumbar chordomas are rare, with long-standing symptoms prior to diagnosis. A high suspicion rate should be present when dealing with a patient who shows pathological nonspecific image studies. MRI images though not pathognomonic, may help reduce diagnostic delay. CT-guided biopsy, with immunohistochemical sample studies, should be thoroughly performed. Treatment can be potentially curative only if aggressive surgical resection is utilized.The authors declare that they have the patient consent.


Adjuvant chemotherapy and radiotherapy were performed. Local control without metastasis was achieved 6 months after excision. This is the first case of intra-articular synovial sarcoma of the hip joint excised via hip arthroscopy. When an intra-articular lesion is identified, malignancies such as synovial sarcoma should be included in the differential diagnosis.Although synovial sarcoma is a relatively common soft tissue sarcoma, primary intra-articular cases are extremely rare. Herein, we report a case of primary intra-articular synovial sarcoma arising from the hip joint, that was initially treated with hip arthroscopy. A 42-year-old male presented with a history of pain in the left hip for 7 years. Radiography and magnetic resonance imaging revealed the primary intra-articular lesion and simple excision with an arthroscopy was performed. Histological findings revealed spindle cell proliferation with abundant psammoma bodies. SS18 gene rearrangement was confirmed by fluorescence  Synovial sarcoma is a relatively common soft tissue sarcoma representing between 2.5% and 10.5% of soft tissue sarcomas .Nevertheless, primary intra-articular sarcomas are extremely rare . AlthougThe patient was a 42-year-old male with a history of continuous pain in his right hip for 7 years without any kind of aggressive sports activities or trauma. He had been walking with a cane for the last year because his pain got worsened. He was referred from the initial hospital for detailed diagnosis and treatment.The radiographs showed calcification around the right femoral neck . The magHistologically, spindle-shaped tumor cells with plump nuclei proliferated with abundant psammomatous calcification . No mitoin situ hybridization using a dual-color break-apart probe covering the SS18 gene revealed rearrangement of the SS18 gene  or synovial osteochondromatosis, which are more common and frequent .Although specific radiological features have not been reported yet , radiogrIn our case, the patient underwent no wide resection but an unplanned resection with arthroscopy. After that, the patient received adjuvant chemotherapy with a first-line combination treatment: ifosfamide and doxorubicin. In addition, radiation therapy was performed after chemotherapy. MRI after chemotherapy and radiation therapy revealed no recurrence of the intra-articular lesion and that the pelvic bone lesion remained stable. In other words, chemotherapy and radiotherapy might be effective. The patient did not have local recurrence or metastatic lesions for 6 months after the end of the treatment.None declared.None.The documents used during the current study are available from the corresponding author on reasonable request.All the report meets ethical guidelines and adheres to the local legal requirements.Written informed consent was obtained from the patient for publication of this case report and any associated images.K.S.


MALAT1-GLI1 gene fusion was considered to be the characteristic molecular alteration of this tumor in previous reports. Herein, we described a 58-year-old man with a mass mainly located in the submucosa of the stomach. Microscopic examination showed a biphasic morphology with the same immunohistochemical phenotype as gastroblastoma. Interestingly, a novel PTCH1::GLI2 fusion rather than MALAT1-GLI1 fusion was detected in the tumor by RNA-based next generation sequencing (NGS). This was the first report that demonstrated a novel PTCH1::GLI2 gene fusion in gastroblastoma, and thus expanded the molecular spectrum of this tumor. The underlying pathogenesis merits further investigation.Gastroblastoma is a rare biphasic tumor of the stomach that generally presents in young patients.  PTCH1::GLI2 gene fusion was found in the tumor cells, instead of the previous reported characteristic MALAT1-GLI1 gene fusion or recent described EWSR1-CTBP1 fusion in gastroblastomas. Thus, our finding expands the molecular spectrum of this extremely rare tumor.Gastroblastoma, first reported by Miettinen et al. in 2009 , is an eAn asymptomatic gastric mass was accidentally found in a 58-year-old man during a routine check-up. The endoscopic ultrasound was performed and a submucosal protuberant mass measuring 1.98 X 1.86cm was detected on the lesser curvature of the gastric body. No treatment was performed at that time. Two months later, repeated endoscopic ultrasound revealed that the mass increased to 2.43 X 1.47cm and showed a hemispherical bulge appearance in the stomach with central ulceration. The mass was uniformly hypoechoic. Enhanced computed tomography scan showed a submucosal mass in the posterior wall of the stomach with suspicion of glomus tumor. The patient underwent endoscopic submucosal dissection.Histological examination showed a well-demarcated tumor centered in the submucosa with focal extension to the mucosa ::GLI2(exon 8) fusion, which was further confirmed by Sanger sequencing (PTCH1 (exon 1) was fused to the 3′ of gene GLI2(exon 8).RNA-sequencing was used to detect known and novel fusion genes across the whole transcriptome. The detailed methods were described in our previous study . It reveGastroblastoma was first described by Miettinen and colleagues in 2009 as a biphasic gastric tumor with variable proportions of epithelial and spindle cells. The reported cases of gastroblastoma had a wide age range at diagnosis, from 9 to 74 years , 7, 8, aPTCH1::GLI2 fusion in our case. However, it requires further verification in more cases. Other gastric neoplasms with biphasic histology should be in the list of differential diagnosis of the present case. Primary synovial sarcoma (SS) of the digestive system has been reported in a few literatures  are the most common mesenchymal neoplasms of the stomach. Some GIST cases may show a biphasic epithelial/spindle cell histology. Negativity for CD34, CD117, and DOG1 by immunohistochemistry can eliminate this diagnosis. In our case, the positive staining for SDHB ruled out the possibility of SDH-deficient GIST as well. Nevertheless, some gastroblastoma cases may show positive CD117 immunoreactivity  signaling pathway. Hh ligands bind to the transmembrane protein receptors Patched  and lift the inhibition of smoothened (SMO) by PTCH, then allowing the transduction of the signal, eventually leading to the activation of GLI which induces the upregulation or downregulation of multiple downstream targets. Hh pathway plays an important role in embryonic development and aberrant activation of Hh pathway may lead to congenital abnormalities as well as tumorigenesis  family consists of GLI1, GLI2 and GLI3, all of which have a highly conserved zinc finger domain and act as important transcription factors in the Hh pathway. GLI1 has been reported to function in variable tumors . Among tPTCH1::GLI1 fusion has been reported recently by Antonescu et al. in a malignant epithelioid neoplasm in the submandibular area .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.


Gastrointestinal duplications can be found in all parts of the gastrointestinal tract. Duplications of the stomach comprise 2-8% of all duplications and are mostly diagnosed during the first year of life. We present a case of a gastric duplication cyst in a 29-year-old female, presenting with epigastric pain and vomiting. Preoperative diagnosis was assumed to be pyloric stenosis. Intraoperatively, a large mass that was attached to the greater curvature was found. Histopathology results were consistent with gastric duplication cyst. Gastrointestinal duplications (GDs) can be found in all parts of the gastrointestinal tract. They are more common in the ileum, followed by esophagus, jejunum, colon, and stomach . DuplicaA 29-year-old female of low socioeconomic status presented with a history of epigastric pain associated with vomiting lasting one month. Her medical history included chronic hepatitis B virus (HBV) infection and previous surgery for duodenal atresia during her first year of birth. Furthermore, the patient was remarkably malnourished, reporting periodic symptoms of upper GI obstruction since her childhood. Esophagogastroscopy was performed with failure to pass the pyloric antrum. Thus, no abnormal mucosa was inspected, and the diagnosis of possible pyloric stenosis was assumed. CT scan reported dilation of the stomach and an associated dilated part of jejunum that compressed the stomach Figures , 2.An exploratory laparoscopy was scheduled, which was converted to an open procedure, due to multiple adhesions. Midline laparotomy was chosen. After extensive adhesiolysis, a large cystic mass arising from the greater curvature with macroscopic characteristics of a diverticulum was found. Wedge resection of the mass was performed, followed by gastrojejunostomy and truncal vagotomy Figures , 4.According to histologic examination, the specimen had a diameter of 11 cm and length of 8.5 cm and its wall was covered with jejunal epithelium, with parts of chronic inflammation and ulceration. It consisted of mucosa and muscular wall that was not in continuity with the resected part of the stomach. The above-mentioned characteristics are consistent with gastric duplication. The patient had an uneventful postoperative recovery and reports no further symptoms six months postoperatively.There are two main types of gastric duplications, the cystic and the tubular type. The cystic type comprises 80% of all GDs and they are not in communication with the gastric lumen, whereas the tubular type has some communication with the gastric lumen and is encountered in the remaining 20% of the cases . The diaPatients usually describe symptoms of abdominal pain, distention, vomiting, weight loss, palpable epigastric masses, and gastrointestinal bleeding among others ,3. GDs aPreoperative diagnosis of such duplications is difficult. CT scan is one of the most useful tools and it shows thick-wall cysts with inner layer enhancement . MRI canEndoscopic ultrasound is another useful tool in the diagnosis of lesions deriving from the gastric wall and can distinguish their origin as follows: inner echogenic mucosal and outer hypoechoic muscle layers are typical of gastric duplications . EndoscoComplications of GDs include infection, bleeding, perforation into the peritoneal cavity, obstruction, and symptomatic compression of adjacent organs ,7,14. MaIn conclusion, gastric duplication in adult patients is a rare condition that is usually misdiagnosed preoperatively. Due to multiple complications and possible malignant transformation, surgical treatment is indicated. In our case, it is unclear whether the symptoms occurred due to gastric duplication or due to chronic pyloric stenosis. Despite that, excision of the duplication cyst and gastric drainage were necessitated. Vagotomy was preferred to life-long proton pump inhibitors treatment considering the patient’s age and socioeconomic status.


Imaging findings of diffuse hemispheric glioma H3 G34-mutant , a new variant of glioma under the World Health Organization classification, have recently been vigorously debated. Here, we report a case of DHG, H3 G34m in which objective assessments of intratumoral microvessels using arterial spin labeling (ASL) were useful for preoperative diagnosis, selection of anti-tumor drugs, and tracking therapeutic responses. The patient was a 34-year-old woman who presented with weakness in the left arm. Preoperative magnetic resonance imaging (MRI) showed no specific findings of hyperintensity on fluid-attenuated inversion recovery imaging and faint enhancement on T1-weighted imaging with contrast media in the tumor. However, ASL showed a convincing finding of high blood flow in the entire tumor, allowing identification of the tumor as malignant glioma. Tumor specimens obtained from biopsy showed that the tumor comprised low-differentiated tumor cells, abundant histiocytes, and highly dense microvessels. Immunohistochemical findings such as positive findings for H3 G34R and p53, and negative findings for IDH-1, ATRX, and OLIG2 led to the diagnosis of DHG, H3 G34m. Based on findings of hyperperfusion on ASL and detection of vascular endothelial growth factor (VEGF), we administered the anti-VEGF antibody bevacizumab. The tumor shrank significantly but remained. However, the residual tumor showed hypoperfusion on ASL, strongly suggesting tumor remission. Objective assessments of blood flow using ASL are useful in clinical practice for patients with DHG, H3 G34 showing non-specific findings on conventional MRI. Diffuse hemispheric glioma, H3 G34-mutant  has been added as a central nervous system tumor in the World Health Organization (WHO) classification of tumors in 2021, as a new variant of pediatric-type diffuse high-grade glioma of grade 4 MRI of DHG, H3 G34m demonstrates non-specific findings of signal-hyperintense areas on T2-weighted imaging (T2WI) and fluid-attenuated inversion recovery (FLAIR) imaging. On gadolinium-enhanced T1-weighted imaging (Gd-T1WI), the frequency of tumor enhancement has been reported as 63%-79.2% T/N) was 3.30.We performed all clinical practices for the present patient in compliance with the precepts established by the Declaration of Helsinki. The patient was a 34-year-old woman with no contributory past or family history. She felt weakness and numbness in the left arm and visited the former hospital. Based on findings from MRI, physicians suspected a high possibility of demyelinating disease and administered one course (3 days) of steroid pulse therapy approximately 3 weeks after the onset of symptoms. However, this proved ineffective and the lesion enlarged along with exacerbation of left hemiparesis and sensory disturbance. The patient was then transferred and admitted to our hospital approximately 2 months after symptoms onset. On admission, the patient had clear consciousness , but presented with severe hemiparesis of the left extremities at stage 2 of the manual muscle test, and sensory disturbance on the left side of the body. Karnofsky performance status (KPS) was 50%. Non-contrast computed tomography (CT) showed a mild hyperdense area in the center of the lesion, but did not identify any obvious calcification A. As finMGMT) could not be examined because specimens obtained from needle biopsy were too small to extract sufficient amounts of DNA.Although CT and conventional MRI on admission only showed nonspecific findings, we were able to differentiate the tumor as malignant glioma with vigorous microvascular proliferation in the entire tumor based on the convincing findings from ASL of hyperperfusion within the entire tumor. For pathological diagnosis, we carried out stereotactic needle biopsy under local anesthesia, targeting a faintly enhanced area of the parietal lobe  was started at first. In addition, we administered bevacizumab  during the period of radiotherapy, because of the findings of high blood flow in the entire tumor on ASL, and the dense microvessels and VEGF expression in histological findings. Two months after starting combined radiochemotherapy, FLAIR showed significant reduction in tumor size, and ASL showed shrinkage of high blood flow areas with a significant decrease in CBFT/N (During the 2 months between biopsy and diagnosis, the FLAIR-hyperintense area rapidly expanded, and high blood flow area enlarged on ASL with an increase in CBFT/N A-D. Togen CBFT/N E-H. Althn CBFT/N I. Howeven CBFT/N I-K. AccoOn conventional MRI, T2/FLAIR imaging shows nonspecific high signals in most patients with DHG, H3 G34m, while patients showing non-enhancement on Gd-T1WI are not uncommon. With regard to the frequency of non-enhancement or only faint enhancement such as in the present case, Puntonet et al. The histological findings of DHG, H3 G34m have remained unclear, because this pathology shows a variety of histological features such as grade 3 astrocytoma MGMT methylation associated with the sensitivity to alkylating agents in patients with DHG, H3 G34m MGMT methylation could not be assessed for this case, the patient showed good therapeutic response and continued progression-free survival for 20 months after starting treatments including bevacizumab. Bevacizumab plays a role in restoration of the disrupted BBB in addition to inhibition of angiogenesis, thereby facilitating transport of other chemotherapeutic agents into the tumor The prognosis of DHG, H3 G34m is better than that of glioblastoma, with a median progression-free survival of 9 months and a median overall survival of 18-22 months Intratumoral calcifications in DHG, H3 G34m can be seen as high signals on T1WI Written informed consent was obtained from the patient for publication of this case report and accompanying images.


The heterogeneity of thoracic wall tumors often represents challenging clinical entities for surgeons due to diagnostic and treatment complexities. The primary tumors, metastases, or direct invasion from intrathoracic structures comprise almost half of all cases on average that are proved to be malignant. Surgery treatment usually leaves large chest defects that require further extensive reconstruction and multimodal management including radiotherapy and chemotherapy. We report a rare case of a giant (30 cm) post-traumatic bleeding thoracic sarcoma treatment in a 70-year-old female. The use of our modified Verneuil technique to close the extensive postoperative skin defect optimized surgical wound management and provided good functional and aesthetic results. Four-year follow-up outcomes after surgical and adjuvant radiation therapy reported a high level of tumor control and showed no evidence of postoperative disease recurrence. Thoracic wall tumors are a rare heterogeneous group of neoplasms accounting for only 5% of all tumors in the thoracic anatomical area . They caThese tumors are rather challenging for physicians because the primary treatment option such as surgery poses a significant and growing risk of morbidity and mortality . The staAnother challenge that surgeons face is removal of giant bleeding tumors. The surgery usually leaves large chest defects that require extensive reconstruction and a multidisciplinary approach .The largest soft-tissue sarcoma of the chest wall described by Davis et al. in 2016 was 27 cm . We pres12, [normal range 3.8–5.2 × 1012] hemoglobin 55 g/L [normal range 120–150 g/L], hematocrit 17.4% [normal range 35%–45%]), hypocoagulation , and elevated liver transaminases levels . Electrocardiography showed sinus tachycardia and left bundle branch block. Other medical examinations were unremarkable. The tumor hemorrhage was controlled by stitching, cottonoids, hemostatic therapy , blood (2 units of 300 ml) and fresh frozen plasma (2 units of 300 ml) transfusions.A 70-year-old female was admitted to the hospital through the emergency department on January 17, 2018, with a giant intensely bleeding thoracic tumor after falling down the stairs and injuring the primary tumor site. According to the patient, the chest wall tumor showed slow growth for 20 years and enlarged significantly at the site of a traumatic injury after the incident. Some years ago, the patient refused surgical treatment plan as her previous healthcare provider had offered her a two-stage surgery with a long recovery period. On admission, her blood pressure was 140/70 mmHg, and her heart rate was 120 bpm. Physical examination revealed a bleeding necrotic tumor mass on the left posterolateral surface of the chest up to 30 cm in diameter and a necrotic wound approximately 10 cm in diameter. The skin immediately surrounding the tumor was hyperemic, moderately edematous, and indurated . The patAfter clinical stabilization, we performed a thoracic and abdominal computer tomography (CT) scan with intravenous contrast media to assess the extent of the tumor. CT demonstrated a giant soft-tissue mass located in the left hemithorax at the T3-L1 vertebra. However, the projection of the dataset acquired on the CT scanner was truncated because the detected mass size extended outside the scan field of view.The tumor measuring 192 mm long and up to 178 mm wide was well-circumscribed, vascularized by dilated and tortuous vessels  and lay Preoperative assessment excluded the possibility of the chest wall invasion. On January 18 after clinical stabilization the tumor was removed with clear surgical margins . Accordiβ-catenin markers were negative. Over 5 mitoses per field of view were detected.The surgically resected specimen showed a solid ulcerous-necrotic mass . HistoloThus, according to morphologic findings the resected tumor was graded as an adult type fibrosarcoma ICD 8810/3. Considering the CD34 expression and the tumor localization, the solitary fibrous tumor had a risk of malignant transformation into a fibrosarcoma.12, hemoglobin: 106 g/L, hematocrit: 30.4%). The patient was discharged in a satisfactory condition and assigned short-course radiotherapy for a total dose of 50 Gy. After 4 years of follow-up, no evidence of recurrence was observed , and American Society of Anesthesiologists score 1  are identified as independent predictors of actual 5-year survival  are diagnosed in 14.3% of cases . The 5-ysurvival . Completsurvival .Determining the preferred technique for large skin defects closure that follow the resection of huge neoplasms is paramount. Chest wall reconstruction is one of the most challenging procedures of the surgery process. It is indicated in a variety of clinical situations, such as rib resection (3 or more ribs), a postoperative soft-tissue defect measured more than 5 cm, the need for adequate chest wall stabilization and prevention of fluid accumulation and fistula formation, airtight closure  and cosmetic appearance improvement. Muscle and musculocutaneous flaps are classically the tissues of choice to cover the wound , 12. TraIn the presented case the patient underwent vascularized free tissue transfer for thoracic chest wall reconstruction. To provide the most adequate result we used the breast soft tissues transfer as the most appropriate reconstruction method. There are several important factors that contributed to the choice of this option as a good alternative. Since the postoperative defect area size was more than 30 cm in diameter, a substantial amount of robust well-vascularized tissue for reconstruction was required to prevent postoperative necrosis of the flap. The vascularization of breasts is abundant and characterized by individual diversity. The main arteries that supply the gland are the internal thoracic artery, the lateral thoracic artery, the thoracoacromial artery, intercostal arteries and some of the vessels that supply nearby muscles . The preBreast mobilization is a surgical technique that implies the use of contralateral mammary gland to close large mastectomy defects . The oriThe postoperative period was uneventful, and the postsurgical scar had no adverse outcomes. The tumor was completely removed, and the patient had no need for additional surgery or reconstructive procedures. Follow-up examinations within four years found no evidence of recurrence.The experience of the giant bleeding thoracic wall chest tumor treatment has shown that the most favorable outcome that can be achieved after surgery is a complete tumor resection in combination with a modified reconstructive surgery method which encompasses mobilization and tissue transfer of the breast towards the ipsilateral side.The closure of skin defects after removal of a large bleeding wall chest tumor using the modification of Verneuil technique optimized postoperative surgical wound and incision healing, provided good functional and aesthetic results, and, therefore, improved the patient's quality of life.


Hepatic portal venous gas is diagnosed via computed tomography due to unusual imaging features. HPVG when linked with pneumatosis intestinalis has a high mortality rate and required urgent intervention. We present a case of a 26‐year‐old young adult with superior mesenteric artery thrombosis who presented with severe abdominal pain. On imaging, HPVG and pneumatosis intestinalis were seen owing to the urgent intervention of the patient. The reliable interpretation of the imaging findings along with quick intervention led to a favorable outcome in our case. Herein, we present a thorough review of the imaging findings of HPVG to make a reliable diagnosis when presented with such a case. HVPG is a rare condition that might be difficult to cure. Is significantly more difficult to treat and has a greater death rate when it is accompanied by intestinal necrosis. In our case, superior mesenteric artery thrombosis led to bowel ischemia. The clinical symptoms of HPVG ranges from diseases that are benign to life‐threatening.While the presence of either HPVG or Pneumatosis Intestinalis (PI) on a CT scan alone does not always indicate that there has been a bowel infarction, the presence of both of these illnesses on a CT scan is strongly linked to this condition, particularly if there is band‐like pneumatosis. It typically denotes irreparable damage and transmural necrosis.2A 26‐year‐old male patient presented to our emergency department with abdominal pain. The pain was gradual in onset, intermittent in nature for a month, which suddenly increased in severity for a day. The pain was not referred to other parts of body. He also had nausea and vomiting associated with it. However, he did not complain of fever. His examination revealed blood pressure to be 130/70 mm hg, heart rate of 100 bpm, respiratory rate of 22 per minute, temperature of 98 °F, and SpO2 93%. Abdominal examination revealed marked abdominal tenderness diffusely. In addition, he had guarding and rigidity present.Laboratory examination showed the following:Following the laboratory examination, a CT scan of the abdomen was done.33.1Noncontrast and contrast CT of abdomen and pelvis were done, which demonstrate linear branching air attenuating areas (HU‐998) in both lobes of liver involving periphery and in the region of portal venous branches, these features are suggestive of portal venous gas Figure . Postcon4Superior mesenteric artery thrombosis leads to the bowel ischemia along with pneumatosis intestinalis and hepatic portal venous gas.5Hepatic portal venous gas (HPVG) is a rarely described form of pneumatosis and refers to gas within the portal vein.Mucosal damage, bowel distension, and sepsis caused by gas‐forming bacteria are the three possible mechanisms of HPVG. A necrotic bowel was present in more than two‐thirds of patients with hepatic portal venous gas. Pneumatosis intestinalis (PI), subserosal and submucosal gas‐filled cysts in the digestive tract, is frequently accompanied by hepatic portal venous gas.HVPG is a rare occurrence, and it is challenging to treat.HVPG is diagnosed radiologically using ultrasound or computed tomography (CT).Treatment of HPVG mostly depends upon the underlying cause. It also depends on the presence or absence of peritonitis or intestinal perforation, as well as the patient's general condition, as they are the primary characteristics that direct clinicians in their therapeutic strategy.6HPVG is not always a sign of a fatal intraabdominal pathology; however, it may be seen in such kind of illness. It can be diagnosed by Ultrasound or CT scan (which is considered as gold standard for its diagnosis). The treatment depends on the underlying pathology as in our case it was due to bowel ischemia resulting from SMA thrombosis. He had to undergo surgical treatment due to the deteriorating clinical status. It is vital for a clinician to recognize when HPVG can be life‐threatening and require intervention.Prakash Dhakal: Supervision. Suraj Sharma: Visualization. Abhishek Sharma: Writing – review and editing. Shailendra Pandey: Writing – review and editing. Sajiva Aryal: Writing – original draft. Seema Bhnadari: Writing – original draft.No funds were received from any institutions or any persons for this case report.There is no conflict of interest among authors.Not applicable.Written informed consent was obtained from the patient to publish the report in accordance with the journal's patient consent policy.


Hamartomas are tumor-like deformities typified by cellular propagation indigenous to the original site, although they display growth arrest without the possibility for further growth. Various hamartomatous oral lesions include hemangiomas, lymphangiomas, nevi, odontomas, Cherubism, etc. Lymphangiomas are benign, developmental hamartomatous entities typified by abnormal proliferation of lymphatic vessels. They are usually congenital, and more than 90% of cases occur by 2 years of age, with a rare occurrence in adults. They have a site affinity for the head and neck, and oral lesions are relatively uncommon. The dorsum of the tongue is the commonest oral site of predilection; however, the lip is a rare site of involvement. Hereby, we present an uncommon case of lymphangioma of the lower lip in a 45-year-old male patient, who reported to our hospital with an asymptomatic lower lip submucosal mass present for the last 3 years. Our case is unique as it occurred on the lower lip of a 45-year-old male. A detailed history and clinical evaluation, ultrasonography, and histopathology confirmed the diagnosis of lymphangioma. Hamartoma is described as a benign, unifocal/multifocal, developmental abnormality, encompassing a blend of cytologically normal mature cells and tissues native to the anatomic site, and exhibits a disordered architectural outline with a preponderance of one of the elements .Lymphangioma refers to hamartomatous entities originating from abnormal lymphatics which cease to drain into other lymphatics or veins, thus causing lymph stagnation resulting in the development of large cyst-like dilated lymphatics , 3. Lymprd of the dorsal aspect of the tongue. However, occasional cases may involve the palate, cheek, gingiva, lips, and alveolar ridges [Owing to the intimate proximity to the primitive jugular lymphatics, most cases occur in the head and neck region 75%), followed by the clavicle and axillary regions (25%) 5%, follo. The orar ridges , 7, 9–13r ridges , 12, 14.Hereby, we present an unusual case of lymphangioma of the lower lip in a 45-year-old male patient. Our case is unique in a few aspects as it manifested age and site variation.A 45-year-old male patient with no known comorbidities reported to our Outpatient Department with a complaint of a mass in the lower lip region for the past 3 years. The initially smaller mass has shown gradual progression, although the lesion has been static for the past 3-4 months. There was a negative history of similar lesions in any other part of the body. However, there was an occasional factitial habit of lip biting. The extraoral examination was unremarkable. On intraoral examination, a solitary, well-delineated nodular mass was noticeable in the submucosa of the lower lip, 2 × 1.5 cm in diameter, extending anteriorly 3 cm away from the mucocutaneous junction, posteriorly blending and obliterating the vestibule, and mesiodistally extending from right canine to the left lateral incisor region. The overlying mucosa was of the same color as the adjacent mucosa, smooth-surfaced with few interspersed areas exhibiting a granular appearance. Palpatory findings were suggestive of a nontender, nonfluctuant mass, soft to firm in consistency, nonpulsatile, and not fixed to the adjacent tissues. Multiple grainy nodules were also felt on palpation. The lesion did not blanch on pressure, and there was no sensory deficit in the lip region. A solitary, sessile, bluish-white, soft, and nontender swelling, 3.5 × 2.5 mm in diameter, was also seen on the right aspect of the vermilion border with mild hyperkeratinization, features in sync with the factitial lip biting. The patient's oral hygiene was fair and class II was seen with 31 Figures . The tooConsidering the anamnesis and clinical appearance of a localized, slow-progressing, smooth-surfaced, well-defined, asymptomatic, freely movable mass located within the lower lip submucosa, the lesion was considered of benign origin. Entities of minor salivary gland origin  and infectious origin (odontogenic/nonodontogenic) and mesenchymal tumors of adipose tissue (lipoma), vascular entities (hemangioma and lymphangioma), smooth muscle lesions (leiomyoma), or neural derivatives  were given a place in the list of differential diagnosis.The lower lip is extremely vulnerable to factitial injury and has a propensity for minor salivary gland tissues; thus, mucocele or salivary duct cysts account for the majority of swellings/lumps in this area. In the present case, the probability of the mass being a mucocele may be negated by its large size (mucoceles larger than 1.5 cm in diameter are extremely infrequent) , age of The possibility of salivary duct cysts is highly unlikely as they characteristically appear as bluish, firm nodular swellings on the floor of the mouth and rarely on the lips .Benign minor salivary gland tumors  primarily involve the upper lip, with infrequent occurrence on the lower lip. Moreover, pleomorphic adenoma is usually seen in younger individuals (<40 years), and canalicular adenoma has an elderly female predilection (>60 years) . These fThe diagnosis of a malignant minor salivary gland tumor (adenoid cystic carcinoma and acinic cell carcinoma) may be ruled out based on the location, well-circumscribed mass, chronic duration, and lack of fixation .Soft tissue infections of the lip (odontogenic/nonodontogenic) may be negated due to the asymptomatic and chronic duration of the lesion with normal dentition. Soft tissue lip abscess is generally ill-defined and fluctuant and exhibits inflammatory signs (pain and erythema) .Lipoma is a nonneoplastic adipose tissue tumor, with an uncommon (1-4%) occurrence in the oral cavity. Lipomas characteristically manifest as a slow-growing, yellow-colored, soft, asymptomatic submucosal mass, with a typical “slip sign,” occurring mostly on the tongue and cheek region .Hemangioma diagnosis may be negated taking into account the patient's age and gender , chronicity of the lesion, noncompressibility, lack of a peculiarly bright red lesion, and a negative diascopy test , 19.Lymphangioma, a benign hamartomatous lesion of the lymphatic system, primarily affects infants and children and involves the tongue, although few lip cases have also been documented . The liprd decade and manifests as a slow-progressing, asymptomatic mass.Leiomyoma, a benign smooth muscle tumor, infrequently occurs in the oral cavity due to the absence of smooth muscle in the region . The lesReactive and neoplastic neural lesions  may also be considered in the present case. The oral traumatic neuroma typically manifests as a nodular lesion, in close proximity to the mental foramen, tongue, or lips. However, the lesions are painful on palpation .Schwannomas (neurilemmomas) are benign neural tumors and manifest as discrete, firm, smooth-surfaced nodular lesions of the same color as the adjacent mucosa. They have a site affinity for the head and neck region, although lip involvement is an extremely rare occurrence , 23.Neurofibroma (NF) is a nerve sheath neoplasm, manifesting as a localized lesion or as a component of the diffuse neurofibromatosis syndrome. However, solitary NF is an extremely unusual occurrence in the oral cavity (6% occurrence) and infrequently affects the lower lip .To summarize, our clinical impression was suggestive of a benign minor salivary gland lesion.With these differentials in mind, the patient was subjected to further investigations. Negative results were obtained on diascopy with a glass slide and fine-needle aspiration (FNA) of the mass that revealed blood-tinged clear fluid resembling lymphatic fluid. The minimally invasive procedure of FNA not only aids in the initial tissue-based diagnosis of salivary gland lesions but also aids in sorting and contemplating the treatment strategy for the patient .Hematologic evaluation including complete blood count, serum calcium, phosphorus, and parathormone levels was within normal limits. A low platelet count (thrombocytopenia) may indicate a risk of bleeding prior to surgical interventions, with a plethora of bleeding symptoms such as bruising, nosebleeds, and, rarely, grievous or fatal bleeding . OrthopaUltrasound was done using a linear probe with special 2D enhancement features including compound and speckle reduction imaging (SRI). The study revealed tiny anechoic cystic lesions with internal septa, measuring approximately 1.9 × 1.0 mm  with adjacent tiny lesions measuring 1.1 × 0.6 mm and 1.7 × 1.5 mm . AnotherMagnetic resonance imaging (MRI) of the lower lip was advised, but the patient denied the investigation due to financial constraints.After taking written consent from the patient, an excisional biopsy was done under local anesthesia. The lesion was enucleated via a blunt horizontal incision of the mucosa with no. 15 blades, taking utmost care not to injure the vital structures. Fine-dissecting scissors and artery forceps were used to excise the lesion. 3-0 vicryl was used to suture the muscle bed, and the lip was approximated and sutured with silk. Grossly, the excised specimen was a nodular mass, firm in consistency, and measured 3 × 2 cm . The enuHistological evaluation of the hematoxylin and eosin-stained sections (10x) reveals an atrophic epithelium beneath which there are numerous dilated lymphatic channels lined by endothelial cells within the papillary and reticular layers of the lamina propria. The large dilated lymphatics just are filled with lymph and infiltrated with inflammatory cell infiltrates, predominantly lymphocytes, neutrophils, and plasma cells. The histopathological features were in coherence with a diagnosis of lymphangioma . HoweverThere exists a diverse pattern in defining lymphangiomas; however, the nonneoplastic proliferation of lymphatic channels delineating a lymphatic hamartoma represents the classical description of lymphangiomas .Lymphangiomas emanate from the sequestration of primordial lymphatic cells. The lymphatic channels persistently accumulate lymph; however, they fail to join with the larger vessels, causing lymphatic obstruction, thus imparting a cystic appearance . They arEmbryologically, lymphangiomas are mostly congenital (50%); the majority of the lesions 90%) manifest by 2 years of age or in rare cases, which may be acquired 0% manife, 28–30. Congenital lymphangiomas can also occur in association with hemangiomas, certain chromosomal aberrations , fetal hydrops, fetal alcohol syndrome, cardiac anomalies, and familial pterygium colli , 13, 31.Our patient was a 45-year-old male with an occasional factitial habit of lip biting. The repeated local trauma in the lower lip region could have triggered the proliferation of lymphatic vessels.rd of the dorsum of the tongue (often resulting in macroglossia) [The close anatomical proximity to the primordial jugular lymphatic sac probably justifies the lymphangioma site predilection for the head and neck region (90% of cases seen in the head and neck region) –11. The glossia) , 13. Othglossia) , 31.The symptoms of adult-onset lymphangioma are atypical and vary considerably, thus, posing a diagnostic dilemma .Lymphangiomas of the lower lip are unusual and account for <1% of the lip biopsies. Lip lesions clinically mimic mucocele, thus causing a diagnostic difficulty , as seenAn extensive literature search was carried out on the Google Scholar and PubMed search engines using the following keywords: lymphangioma, lymphatic malformation, oral cavity, lip, and lower lip. Case reports published in the English language up to December 2021 were thoroughly searched. This extensive bibliographic research finally resulted in 16 case reports, after excluding studies that were duplicates, not case reports, and those which were not in the English language.The parameters extracted from these cases are summarized in The reported cases of lip lymphangiomas are summarized in The lesion extent directs the clinical appearance of lymphangiomas , 11, 14.Our case presented as a solitary, slow-growing, well-circumscribed, asymptomatic, soft-firm, nonblanching, nodular submucosal mass in the lower lip region with normal overlying mucosa, although a few interspersed areas exhibited a granular appearance. A solitary, sessile, bluish-white, soft, and nontender swelling, 3.5x 2.5 mm in diameter, was also seen (in proximity to the mass) on the right aspect of the lip vermilion border with mild keratinization.Several detrimental effects such as breathing and speech difficulty, pain, tongue protrusion, or jaw anomalies may be seen with deep lesions owing to the impingement on the contiguous anatomical structures , 13, 27.Secondary infection may predispose to Ludwig's angina. The postsurgical sequalae may include bleeding, seroma formation, repeated episodes of cellulitis, and leakage of lymphatic fluid .Several hypotheses have been recommended for the etiopathogenesis of lymphangiomas. The first hypotheses propose that congenital lymphangiomas result from sequestration of primordial lymphatics; however, they fail to connect to the main lymphatic vessels or veins . The secHistopathologically, lymphangiomas consist of dilated lymphatic vessels with one or two endothelial layers, with or without an adventitial layer. Based on the location and contiguous structures, these dilated lymphatic vessels are of variable sizes, thus forming the basis of classification. Cavernous lymphangiomas commonly occur in regions with a rich supply of denser connective tissue and skeletal muscle, causing confined lesions, e.g., oral cavity . This is contrary to cystic hygromas, which develop in loose adipose tissue sites resulting in a diffuse spread and large multicystic lesions , 34.The microscopic findings in our case were incoherent with the published literature.Most lymphangiomas are diagnosed clinically based on the characteristic manifestations. However, excision and histological assessment, together with diagnostic radiographic aids facilitate the confirmatory diagnosis of lymphangiomas with bizarre clinical characteristics , 34.Ultrasound (USG) is an essential diagnostic aid in assessing the site, shape, size, and extent of lesions. Ultrasound generally exhibits multicystic lesions with internal septations, and the color-flow Doppler depicts the avascular nature of lymphangiomas, thus, not only distinguishing vascular malformations from hemangiomas but also identifying the various vascular patterns of such entities.In our case, the USG findings were suggestive of multiple anechoic cystic lesions with internal septa on the right and left sides of the lower lip. The color Doppler was suggestive of an avascular anechoic lesion.However, computed tomography (CT) and magnetic resonance imaging (MRI) are superior to ultrasound in describing the relationship with contiguous anatomical structures and demarcating the lesion extent , 29.Magnetic resonance imaging (MRI) of the lower lip was advised, but the patient denied the investigation due to financial constraints.Mostly, lymphangiomas are benign in origin; however, they may require surgical treatment due to their infiltrative pattern causing larger lesions to encroach the adjoining vital structures, resulting in cosmetic, functional, and life-threatening sequelae , 7.The treatment strategies should focus on restoring the functions, averting any potential complications , and attaining adequate cosmetic satisfaction . A wide Sclerotherapy with 25% dextrose, hypertonic saline, bleomycin, and picibanil (OK-432) is considered deemed for recurrent, unresectable/surgically demanding lesions . SclerosRecently published reports have also documented the successful management of tongue lymphangiomas by nonablative long-pulsed Nd:Yag lasers and constitutional homoeopathic medicine Tuberculinum, respectively , 41.Surgical extirpation has an accompanying high recurrence rate (10-39%), primarily due to the deeper permeation of the lesion or incomplete surgical removal. Preoperative intralesional administration of sclerosing agents may prove beneficial in such cases .In our case, surgical enucleation of the mass was done using a blunt horizontal incision under local anesthesia.Lymphangiomas are common entities in the pediatric age group and are rarely encountered in adulthood. They may be frequently misdiagnosed owing to the bizarre clinic radiographic manifestations, and histopathologic evaluation provides the confirmatory diagnosis. Lymphangiomas uncommonly involve the oral cavity, with a rare occurrence in the lower lip. Lymphangiomas should always be given a place in the differential diagnosis of a localized submucosal mass of the lower lip.


Masson's tumor, also known as ‘Masson's vegetated intravascular hemangioendothelioma’, is a papillary hyperplasia of the endothelial vascular cells that generally develops in medium-sized veins, but may also be observed in all-sized veins and, less frequently, in arteries.The publication of clinical data with respect of the laws concerning privacy and was approved by the Ethics Committee of the University Hospital, and the patient consented to the publication of the clinical data regarding this case report.A 43 year-old woman was referred to our Department presenting with a 4 years history of right parotid swelling. The tumor was solid in consistency, had slowly increased in size until 2 cm in its greatest dimension and was mildly painful. The rest of the clinical examination was unremarkable.Contrast-enhanced magnetic resonance imaging (MRI) showed a multilobulated lesion of approximately 2.3 cm in diameter within the superficial lobe of the parotid gland. The lesion was elongated and composed of tubular, tortuous and ectasic structures with suggestive characteristics of a hyper vascularized benign lesion .Figure 1® – Germany) with a focal length of 250 mm and continuous intraoperative facial nerve monitoring . The deeper aspect of the mass was strictly adhering to the peripheral zygomatic branches of the facial nerve and required an extremely precise microscopic dissection to avoid accidental rupture of the lesion and major bleeding. Histological examination showed a major salivary gland (5 cm × 3 cm × 1 cm) with a wide vascular cavity, containing an organizing thrombus and a small papilla protruding into the lumen composed of a single layer of endothelial cells; these cells appeared swollen, without significant pleomorphic or mitotic figures and surrounding a core rich in collagen , a mixed type (secondary or reactive) due to focal changes in a pre-existing vascular lesion  and, rarely, a third type resulting from the organization of a hematoma in an extra vascular location.Head and neck Masson's tumors are found more frequently in the lateral region of the neck, where the most common symptom is a tender or solid slow-growing swelling that can rapidly increase in size, due to the bleeding inside the lesion.Parotid Masson's tumors are extremely rare: In 1982, Corio et al. reported a Masson's tumor of the parotid, but no clinical or therapeutic data were detailed in their descriptionDifferential diagnosis includes all benign and malignant soft-tissue tumors located in the anatomic region where the Masson's tumor develops; the case reported presented more similarly to typical benign parotid tumors.Radiologic diagnosis can be challenging: MRI is equivocal in the diagnosis because of the amount of blood vessels and thrombi usually present within the lesion and a radiologic similarity to the low-grade angiosarcoma.Parotid Masson's tumor is a slow-growing benign lesion adhering to the surrounding salivary gland parenchyma; therefore the surgical removal through sub-total or total parotidectomy is considered the gold standard treatment. In our case the main challenge was due to the inflammatory behavior of the lesion, requiring a fine dissection of the branches of the nerve; the use of the operative microscope associated with the intraoperative nerve monitoring allowed the anatomic preservation of all the branches of the facial nerve, avoiding postoperative weakness of the nerve.Pathological analysis of the case demonstrated an intravascular papillary growth composed of endothelial hyperplasia and a central thrombotic area . EndotheThe prognosis of Masson's tumor is excellent, with extremely low recurrence rate after complete excision.The authors declare no conflicts of interest.


Pilomatricomas are frequently misdiagnosed benign neoplasms of the skin derived from hair matrix cells. Pilomatricomas may undergo calcification and ossification although the latter is rare and poorly documented, with only eight cases reported since 2006. We present a case of pilomatricoma with osseous metaplasia arising from the scalp in an 87-year-old female patient. She was referred by her general practitioner via the two-week cancer referral pathway, for a suspicious lesion. On examination, there was a 2 x 1cm nodule, with protruding hardened yellow material, on the right side of the patient’s occipital scalp. The lump was hard, non-tender and had been present for 17 years. The surrounding area was bleeding and slightly ulcerated. A clinical diagnosis of a ruptured epidermal cyst was made, and the patient was prepared for excision under local anaesthesia. The excised lesion of 23 x 18 x 10mm with 22 x 9mm of skin was sent for histology. This revealed a partially ulcerated dermal lesion composed of islands of keratin with ‘ghost cell’ outlines. Foreign body granulomas, transition to mature lamellar bone, and foci of calcification were noted. There were no definite populations of basaloid cells and features of malignancy were not seen. These findings are consistent with benign pilomatricoma with osseous metaplasia. The patient was discharged 4 weeks later with satisfactory wound healing. Differentiating this tumour from other commonly encountered benign masses remains a challenge, as seen in this case which was initially misdiagnosed as a ruptured epidermal cyst. Pilomatricoma, previously known as calcifying epithelioma of Malherbe, is a frequently misdiagnosed benign neoplasm of the skin associated with hair follicles . The prePilomatricomas were first described in 1880 by Malherbe and Chenantis and most frequently appear as asymptomatic, firm, solitary nodules ,4. The tAn 87-year-old female patient was referred for a suspicious lesion by her general practitioner via the two-week cancer referral pathway. The patient presented to the dermatology department with a 17-year history of a lump that had recently become harder, on the right side of her scalp  mediates fibroblast metaplasia to form osteoblasts in late-stage pilomatricomas . PilomatDifferentiating this tumour from other commonly encountered benign masses remains a challenge, as seen in this case which was initially misdiagnosed as a ruptured epidermal cyst. The diffuse yellow colour of the tumour seen in this case is not common in pilomatricomas. Characteristic red-blue discolouration over the lesion, associated with haemorrhage of the pilomatricomas , was notIn February 2020, Bharti et al.  performePilomatricoma with osseous metaplasia is rare and should be considered as a differential diagnosis in patients presenting with circumscribed, firm, non-tender nodules. The condition is often misdiagnosed, and correct diagnosis is often only established after excision and histological examination.