To report a case of myomatous erythrocytosis syndrome (MES) with an extra-uterine manifestation.A 43-year-old woman presented with progressive abdominal distension and rapid enlargement of a pelvic mass. Upon survey, a high-level of hemoglobin (19.0 g/dl) was documented. The initial impression was an ovarian malignancy, but uterine sarcoma could not be ruled out because of its rapid growth. However, during exploratory laparotomy, the pelvic mass was found to be a 31 cm broad ligament leiomyoma; which is extremely rare for its size and location. The specimen was further studied immunohistochemically, which revealed excessive expressions of erythropoietin and erythropoietin receptors in addition to the diffusely matured blood vessels in the myoma tissue. The patient’s hemoglobin level resumed to normal three months post-surgery. The diagnosis of MES was confirmed both clinically and histologically.A correct preoperative diagnosis is challenging when MES manifests as an extrauterine mass. The coexistence of MES should be considered in the management of all leiomyoma with polycythemia, regardless of locations. Erythropoietin (Epo) is produced by interstitial fibroblasts in the kidney. Polycythemia, defined as an increased hemoglobin level >16 g/dl) or increased hematocrit (>48%) in women, can result from tumor-induced ectopic Epo production. Myomatous erythrocytosis syndrome (MES), characterized by erythrocytosis associated with uterine leiomyoma, is a rare condition with less than 60 cases reported till date 6 g/dl or. Here, wA 43 year old Taiwanese woman, gravida 2, para 2, reported no remarkable surgical and medical history prior to this event. She had been experiencing progressive abdominal distension and a body weight loss from 57 to 52 kg over the last six months. Upon physical examination, the abdominal mass appeared enlarged similar to seven gestational months. Computerized tomography scan revealed a 30 cm heterogeneous mass occupying the entire pelvic space, which was too large for anatomic distinction. The initial laboratory survey revealed a normal CA-125 level, a slightly elevated level of lactate dehydrogenase at 292 U/L (reference range: 120–240 U/L), and an elevated hemoglobin level of 19.0 g/dl. The first impression was an ovarian tumor, but a uterine sarcoma was not ruled out owing to its rapid growth. Exploratory laparotomy was subsequently arranged. During surgery, the pelvic mass was found to have originated from the right broad ligament, while the right ovary had a 3 cm dermoid cyst . Total h3; and under histological examination, a leiomyoma originating from the broad ligament was confirmed. To clarify the association between the myomatous tissue and erythrocytosis, immunohistochemical (IHC) staining was performed with antibodies against erythropoietin  and erythropoietin receptor (Epo-R) . A diffuse Epo expression with moderate to strong staining intensity appeared over the myomatous tissue, but the staining was negative for the endothelial cells , and continued to be within the normal range. Therefore, the diagnosis of MES associated with a broad ligament leiomyoma was confirmed both clinically and histologically.In 1957, Fleming et al.  proposedOn gross inspection, specimens from the patient revealed only small extent of degeneration, which was somewhat an unusual feature considering its large size. As a myoma outgrows its blood supply, it usually undergoes degenerative changes, with hyaline degeneration being the most common manifestation in 60% of the reported cases . TherefoIn our case, IHC staining revealed diffuse Epo expression throughout the myomatous tissue; however, the endothelial cells were not similarly stained. This finding provides direct evidence that ectopic Epo was produced by the myomatous cells rather than by endothelial cells. In addition, H&E staining demonstrated widespread, highly-matured blood vessels in the myomatous tissue, which was compatible with a vessel maturity score of 5, as defined in previous studies . These fvia Epo-R, which activates the mitogenic pathway, EpoR-JAK2-STAT5, and promotes paracrine angiogenesis (Epo and Epo-R are present in malignant cells of the cervix, endometrium and ovary. The tumor and vascular endothelial cells are sites responsive to Epo . In tumoogenesis . This loogenesis . StudiesIn the present case, reduction of the risk of polycythemia-associated thromboembolism was attempted by prophylactic administration of 40 mg enoxaparin prior to the surgery. Unosawa et al.  reportedBroad ligament is the most common site for extra-uterine myoma, with an incidence of ≤1% . Only twThe incidence of MES is surprisingly low when compared with the high prevalence of uterine leiomyoma. With the report of Epo mRNA present in 95% of leiomyomata . In addiIn conclusion, a correct preoperative diagnosis of MES is challenging when it manifests as an extra-uterine mass. As MES usually mimics an ovarian malignancy, a misdiagnosis can lead to unnecessary surgical procedures or even chemotherapy. The risk of polycythemia associated thrombosis also necessitates pre-operative prophylaxis. The coexistence of MES should be considered in the management of all leiomyoma with polycythemia, regardless of its location.


A male patient in his 70s, who had undergone an abdominoperineal (A1) resection for rectal cancer 20 years prior, presented with hemoptysis. Imaging studies revealed distant lung recurrence, with no evidence of local recurrence. Biopsy revealed an adenocarcinoma, possibly of rectal origin. Immunohistochemical markers were suggestive of rectal cancer metastasis. However, carcinoembryonic antigen (CEA) levels were normal, and colonoscopy did not reveal any metachronous lesions. Curative left upper lobe resection was performed via posterolateral thoracotomy. The patient’s recovery was uneventful. Colorectal cancer is the third most common cancer in the world and the second most common cancer-causing death as per the GLOBOCAN 2020 . DependiA male patient in his 70s was diagnosed with rectal cancer and underwent abdominoperineal resection 20 years prior. He initially presented with painless rectal bleeding, which was investigated using facilities available at that time. Colonoscopy revealed a malignant lesion located 5 cm from the anal verge. No other synchronous lesions were observed. Histological findings were suggestive of an infiltrative, moderately differentiated adenocarcinoma with many villous structures, and grade 3 dysplasia. Chest and liver imaging did not reveal any metastatic lesions. Abdominoperineal resection was performed, and a biopsy revealed complete excision of the tumor with adequate margins. The patient subsequently underwent six cycles of adjuvant chemotherapy with folinic acid, fluorouracil, and oxaliplatin (FOLFOX). He was managed in accordance with the National Comprehensive Cancer Network (NCCN) guidelines, and was followed up with regular imaging, assessment of CEA levels, and colonoscopy for up to five years.After 20 years, the patient presented with a one-week history of hemoptysis. He was examined for possible causes of hemoptysis at the respiratory unit. He had a dry cough, pleuritic chest pain, no fever, no history of contact with a patient with tuberculosis, no weight loss, and no loss of appetite. He had no history of cigarette smoking or alcohol consumption but had occupational exposure to coal smoke. The patient had no family history of lung or rectal cancer. Physical examination did not elicit icterus, cervical lymphadenopathy, or lung signs.An initial chest radiograph revealed a lesion in the upper lobe of the left lung Figure . CEA wasThe case was discussed in a multidisciplinary team meeting and two options were suggested. The first was metastasectomy and adjuvant chemotherapy, while the second was neoadjuvant chemotherapy followed by surgery. The options were discussed with the patient, and he chose the first option. A left upper lobectomy was planned using an open surgical approach. Fitness for surgery was assessed preoperatively. A posterolateral thoracotomy was performed along the fifth intercostal space. Pleural reflections and the lungs were identified, after which the pleural reflections were divided. The upper lobes of the pulmonary artery, bronchus, and pulmonary veins were identified and ligated. The anterior and posterior ends of the oblique fissure were divided using a linear stapler. The upper lobe along with the tumour was then removed Figure . An intePulmonary resection in patients with colorectal cancer and lung metastasis has survival benefits -8. DFI, DFI is defined as the time interval between colonic resection and lung resection. The median DFI described in the literature ranges from 18 to 37.5 months with the longest DFI being 260 months -7. In thPatients with normal pre-thoracotomy CEA levels are considered to have a better prognosis than those with elevated levels -7. In thIt is recommended that after performing a colonoscopy in the first year following curative surgery, the next colonoscopies should be performed at three years and five years postoperatively. Subsequent colonoscopies should be performed at five-year intervals . The NCCThere are rare presentations of distant lung recurrence following curative surgery for rectal cancer, even after years. This should be considered during the follow-up management of patients with rectal cancer. Pulmonary resection in colorectal cancer with lung metastasis has survival benefits for the patient. DFI, number of lung lesions, and the pre-thoracotomy CEA levels are important prognostic factors.


The patient was a male neonate, and a prenatal ultrasound had detected a right lung mass. He was born at term and after delivery had tachypnea and feeding difficulties. A chest x-ray and a computed tomography (CT) scan revealed a large mass in the right chest with compression on the right lung after birth. We initially considered congenital pulmonary airway malformation (CPAM). After conservative treatment, his respiratory symptoms worsened gradually, and he required continuous supplemental oxygen. The symptoms could not be relieved by puncturing due to a postnatal ultrasound having shown a mass with anechoic microcystic spaces. He therefore underwent an emergency thoracotomy and lobectomy at 14 days of age. The pathology was consistent with fetal lung interstitial tumor (FLIT). The patient remained healthy at the three-month follow-up. We reviewed the literature on FLIT and found that, to date, 23 cases have been reported worldwide. Fetal lung interstitial tumor (FLIT) is a newly-defined disease first reported by Dishop et al. , 2. ReadMisdiagnosis may increase clinical risk because the progression and treatment of FLIT differs from other conditions. Dishop et al. report that, in one case, FLIT was misdiagnosed as airway obstruction and did not receive surgery until doctors observed that the mass had increased . Prompt The patient was a male neonate . A prenatal ultrasound, at 33 weeks and 2 days of gestation, showed a solid, minimally hyperechoic, right-sided lung mass with cystic components, which suggested CPAM . The CPA3) with anechoic cystic spaces  is easily misdiagnosed, especially as being CPAM , 4. In oOf the 23 reported cases, four were detected before birth, 18 were detected when symptoms developed after birth, and one was not described . These fA correct FLIT diagnosis can enable patients to receive timely and proper treatment. In the case of fetal edema, doctors can use hormones to try and relieve it. Phillips et al. report a case treated with betamethasone but that where polyhydramnios remained . DoctorsMost FLIT cases received a lobectomy . In our A pathological diagnosis cannot be confirmed by H&E staining alone, but with further specialist staining –18. To dFLIT is easily misdiagnosed. Prenatal ultrasound can aid diagnosis and should be performed regularly. An increasing mass volume in later gestation can indicate FLIT. After birth, feeding difficulties is a relatively specific symptom. In FLIT, a postnatal ultrasound shows a mass with anechoic microcystic spaces. CT scans usually show a mass with uniform density in FLIT, but a mass with low-density cystic areas in CPAM. Correct diagnosis can enable patients to receive timely and proper treatment. FLIT should be excised as early as possible, and most surgeons perform lobectomy. During surgery, surgeons should carefully monitor airway anatomy in order to avoid airway damage.


Lichen planus pemphigoid (LPP) is a rare autoimmune bullous dermatosis, although it is frequently idiopathic, the induced form is rare and there are few inducing drugs. We report a case of LPP induced by a gliclazide. A 68-year-old female patient with type 2 diabetes on gliclazide for three months presented with an eight-week history of generalized erythematous-papular eruption. Then she developed blisters on her forearms with oral mucosa involvement. A diagnosis of gliclazide-induced LPP was made based on a skin biopsy and imputability. The patient was treated with systemic corticosteroid with an improvement. LPP is a rare entity; its diagnosis is a challenge as it represents an overlap between lichen planus and bullous pemphigoid. Lichen planus pemphigoid (LPP) is an uncommon acquired autoimmune bullous dermatosis marked by the coexistence of tense bullae and lichen planus lesions . LPP is A 68-year-old female patient with type 2 diabetes on gliclazide for three months, without any other medication, developed pruritic papules on her trunk and extremities for two months, followed six weeks later by bullous lesions on her forearms. A clinical examination found generalized polygonal and violaceous, papules with Whickham streaks on the trunk, back, abdomen, legs, and thigh Figure , topped Examination of the oral mucous found a whitish lichenoid network Figure . A genitHistopathological examination of the papule found an orthokeratotic epidermis, with exocytosis of neutrophils and apoptotic bodies. The dermis was the site of a lichenoid inflammatory infiltrate. The histology of the bullous found a subepidermal blister with inflammatory infiltrates made of eosinophil in the dermis. Direct immunofluorescence showed a junctional deposition of C3 and IgG. The blood count showed 780-element hyper eosinophilia. The pharmacovigilance survey found an accountability score according to the French I3B4 method. Gliclazide was discontinued and replaced with insulin; the patient received oral prednisone 0.5 mg/kg because of the extent of the lesions with improvement.LPP is a rare and acquired autoimmune bullous dermatosis. It was first described by Kaposi  in 1982 The histopathology of a bullous lesion has the typical pattern of bullous pemphigoid (BP): subepidermal separation, with multiple eosinophils in the vesicular fluid and an eosinophilic infiltrate, while the histopathology of a lichenoid lesion has the typical characteristics of lichen planus . The etiPharmacovigilance databases are another precious tool to detect drugs that may cause the development of LPP. Gliclazide is a second-generation sulphonylurea oral hypoglycaemic drug, used widely in the treatment of type 2 diabetes in the population , includiAccording to the literature, angiotensin-converting enzyme inhibitors are the most prone to LPP, while oral antidiabetics recognized by the lack of cutaneous side effects are increasingly inducing LPP. To our knowledge, this is the second case report of oral antidiabetic associated with the development of LPP. Gliptin is known to cause BP. Its pathophysiological mechanism and the report of our case may open the debate about whether LPP is really a variant of BP or if it should be considered a completely different entity.


With the development of laparoscopic techniques and the broad clinical application of various anastomotic types, anal-preserving low anterior rectal resection and ultra-low anterior rectal resection have been popularized. Some patients with rectal cancer have retained their anus and improved their quality of life. Nevertheless, the incidence of postoperative anastomotic stenosis remains high, and anastomotic occlusion is even rarer.We report a case of anastomotic occlusion in a patient with rectal cancer, which occurred after undergoing laparoscopic low anterior rectal resection + prophylactic terminal ileal fistulation at our department. Under endoscopy, we used a small guidewire to break through the occluded anastomosis, thereby finding the lacuna. After endoscopic balloon dilation, digital anal dilatation, and continuous dilator-assisted dilation, the desired efficacy was achieved, ultimately recovering ileal stoma. Postoperative follow-up condition was generally acceptable, without symptoms like abdominal pain, bloating, or difficulty in defecation.Numerous factors cause postoperative anastomotic stenosis in patients with rectal cancer. Complete occlusion of anastomosis occurs relatively rare in clinical practice, and is challenging to treat. This case was our first attempt to remove the anastomotic occlusion successfully, which avoided re-operation or pain from the permanent fistula. With the application and popularization of laparoscopic techniques and anastomosis, laparoscopic surgery has become the preferred treatment for rectal cancer. It has the advantages of a clear surgical field, better convenience than open surgery, low invasiveness and fast recovery. Anastomotic fistula and stenosis are major complications following colorectal surgery , 2. The A 53-year-old female patient was hospitalized at our department on August 21, 2020 because of the “rectal mass found 7 days ago.” Digital anal examination findings (chest-knee position): a hard mass of approximately 3 × 3 cm in size was palpable at the 7–9 o’clock orientation 4 cm apart from the anus, with no tenderness and moderate mobility. No blood stain was detected when exiting the finger cot, and the tumor markers were normal. Abdominal CT and pelvic MRI findings: space-occupying lesions in the lower rectum. Preoperative diagnosis: “moderately differentiated adenocarcinoma of the lower rectum(T3N0M0)”. On August 31, 2020, the patient received “laparoscopic low anterior rectal resection + prophylactic terminal ileal fistulation" under general anesthesia. After the surgery, the anastomosis was unobstructed based on digital detection, about 2.8 cm in diameter and 3 cm away from the anus, with circular anastomotic nails palpable. Postoperative pathological diagnosis: moderately differentiated adenocarcinoma of the lower rectum. Good postoperative recovery was achieved without developing complications such as anastomotic leakage, stenosis, or bleeding. The patient refused chemotherapy due to personal reasons. She was told to receive recovery of small intestinal stoma 3 months later, during which regular follow-up visits were required.After hospital discharge, the patient did not return for re-examination. On December 1, 2020, she visited our department and requested a small intestinal stoma recovery. Digital anal examination revealed a blind cavity at the rectal end, with undetectable anastomosis and palpable anastomotic nails. Colonoscopy and abdominal CT showed occluded anastomosis Fig. . Later, Anastomotic stenosis (AS) is one of the late complications following rectal cancer surgery. Very few patients develop anastomotic occlusion. Symptoms of AS usually include difficulty in defecation, bloating, and anal pain . Causes There are many treatments strategies for AS, including mechanical dilation, endoscopic therapy and transabdominal surgery –11. ThesThe occurrence of AS is associated with numerous factors, where prevention should be emphasized. Once discovered, early treatment should be given. Complete occlusion is a rare condition that is difficult to manage. Improper management may exacerbate the patients' condition and economic burden. From simple to difficult and from non-invasive to invasive, the therapeutic strategy should depend on individual conditions –19. Wher


Bifid mandibular condyle (BMC) is splitting the mandibular condyle into two separate articular surfaces. The etiology is poorly understood, but trauma and developmental issues are currently the most cited causes. Though most often asymptomatic, occasionally, this condition may cause the development of jaw pain, clicking, and restriction of motion. We present a rare case of a patient who developed unilateral ankylosis of the temporomandibular joint (TMJ) secondary to BMC in the absence of trauma or infection. The ankylosis developed due to abnormal biomechanical forces and degenerative arthritis secondary to the abnormal articulation of the TMJ caused due to BMC. CT imaging is the best modality to evaluate the bony anatomy of the TMJ. It is essential to consider BMC as a cause of TMJ pathology, as management is primarily surgical in nature. Bifid mandibular condyle (BMC) is a rare skeletal abnormality that involves the separation of the mandibular articular condyle into two articular surfaces. These surfaces divide either anteroposteriorly or mediolaterally -3. PreviA nine-year-old boy presented with a six-month history of progressive jaw deviation and difficulty opening his mouth, with a sensation of the left side of his jaw feeling “stuck.” There was no reported recent or remote history of trauma, surgery, or jaw infection. In addition, the patient had no documented history of metabolic or genetic skeletal abnormalities and was otherwise healthy.A significant leftward deviation of the mandible was noted on physical examination relative to the maxilla and skull base. A significant overbite was also appreciated. The facial movement was normal, but the jaw opening was limited. No facial masses or cervical lymphadenopathy were noted. A CT of the maxillofacial region with and without contrast was obtained to further evaluate this condition. The contrast was administered to confidently rule out infection, despite the absence of clinical signs such as fever, erythema, and edema.CT imaging demonstrated an abnormal left TMJ with two sites of articulation oriented medially and laterally. The medial component of the condyle appeared to articulate with the temporal process of the zygomatic bone with an abnormally flattened appearance. The medial articulation showed no joint narrowing, erosion, or lytic lesions Figures -2A.The lateral articulation appeared to have an osseous bridge extending between the lateral mandibular ramus and the lateral process of the temporal fossa. This “joint” space was narrow, with significant erosive changes in both opposing surfaces of the articulation. There was no evidence of periosteal reaction, involucrum, or additional lesions. No soft tissue changes, fat stranding, edema, or fluid collections were appreciated Figures -2B. The BMC is a rare anatomic abnormality with an estimated prevalence of 0.018%-1.82% ,7. Some BMC is often found incidentally on panoramic dental radiographs or head and neck imaging ,10. CT iThere have only been about 50 reported cases of BMC with associated ankylosis ,15-19. AManagement of mandibular ankylosis is typically surgical in nature. Prior successful treatment has been achieved with condylectomy, bilateral coronectomy, and genioplasty to correct chin deviation . Mild caIn our case, BMC presented with rapidly progressive symptoms and ankylosis in the absence of trauma or infection. Thus, it is essential to consider mechanical changes as a cause of ankylosis in patients with BMC. Though BMC is rare and continues to be poorly understood, it should be considered when there is a pathology of the TMJ. CT imaging should be obtained to evaluate the bony anatomy of the TMJ if pathology is suspected. Regarding management, surgical evaluation should be obtained for patients with symptomatic BMC. There are no clear guidelines to direct either surgical or medical management. However, a decision should be made based on the severity of the patient’s symptoms and their impact on quality of life.


Primary hepatic extranodal marginal zone B-cell mucosa-associated lymphoid tissue (MALT) lymphoma is very rare, so it is difficult to diagnose preoperatively. And there is no established treatment for hepatic MALT lymphoma. We report herein a case of primary hepatic MALT lymphoma treated by laparoscopic partial hepatectomy, and discuss the usefulness of laparoscopic hepatectomy for a rare liver tumor.This patient was a woman in her 60s, who was diagnosed preoperatively as having synchronous liver metastasis from sigmoid colon cancer; therefore, laparoscopic partial hepatectomy was performed. She had a good course after the operation and was discharged on postoperative day 12. However, she was diagnosed pathologically as having primary hepatic MALT lymphoma. A bone marrow biopsy was also performed, and then she was finally diagnosed as having limited-stage primary hepatic MALT lymphoma. She received no postoperative treatment and showed no recurrence for 4 years postoperatively.We experienced the good result of the patient with limited-stage primary MALT lymphoma treated by laparoscopic partial hepatectomy. Liver tumors are sometimes misdiagnosed by imaging examinations alone. Laparoscopic hepatectomy has been widespread recently as a minimally invasive procedure, and it may be useful for both diagnosis and treatment. One of the indolent lymphomas is an extranodal marginal zone B-cell mucosa-associated lymphoid tissue (MALT) lymphoma. MALT lymphoma occurs most frequently in the stomach , but it This report presents a case of primary hepatic MALT lymphoma treated by laparoscopic partial hepatectomy. The patient had been initially diagnosed as having synchronous liver metastasis from sigmoid colon cancer, but the pathological diagnosis was primary hepatic MALT lymphoma postoperatively. In this case, it was not until the resection of the liver tumor that it was diagnosed, and the treatment was completed only with laparoscopic hepatectomy, which was minimally invasive.18F-fluorodeoxyglucose-positron emission tomography (FDG-PET). MDCT showed a low-density nodule during the arterial phase , pT3(SS), med, INFb, ly0, v1, pN0(0/16), EX/ND(-), PN1a, pPM0 (90 mm), pDM0 (50 mm), pRM0. She received postoperative systemic chemotherapy. Then, no new lesion occurred in the liver. She preferred to undergo laparoscopic hepatectomy and was referred to our hospital. She tested negative for serum marker of hepatitis B and C virus infection. Additionally, several types of imaging examinations were performed: multidetector computed tomography (MDCT), Gd-EOB-DTPA-enhanced magnetic resonance imaging (EOB-MRI), and The trocar was placed, with no adhesions and no ascites in the abdominal cavity. The liver was examined intraoperatively by contrast-enhanced ultrasonography with perfluorobutane microbubbles Fig. a, and thThe patient had a good postoperative course, and she was discharged on postoperative day 12.The pathological findings were as follows. The nodule in the liver parenchyma was composed of dense lymphoplasmacytic proliferation, and scattered lymph follicles were surrounded by intermediate-sized B cells with clear cytoplasm, suggesting marginal zone/monocytoid B cell differentiation Fig. a. Those Primary hepatic MALT lymphoma is quite rare and was first reported by Peter G. Isaacson in 1995 . It accoLiver tumors are diagnosed mainly by imaging examinations. At our hospital, we usually perform MDCT, EOB-MRI, and FDG-PET. This case showed ring enhancement during the delayed phase on MDCT and restricted diffusion on EOB-MRI. However, it did not show arterial phase enhancement or the vessel penetration sign. From the above findings, liver metastasis from sigmoid colon cancer or primary intrahepatic cholangiocarcinoma was diagnosed, and, therefore, surgical resection was performed. Primary hepatic MALT lymphomas were reported to be characterized by arterial phase enhancement, restricted diffusion, the vessel penetration sign, and, more specifically, “speckled enhancement” in the hepatobiliary phase of EOB-MRI. “Speckled enhancement” refers to punctate positive enhancement within the low-signal intensity lesions on the hepatobiliary phase of EOB-MRI . HoweverHelicobacter pylori, so Helicobacter pylori eradication therapy should be given to all patients with gastric MALT lymphoma, irrespective of stage [Treatment for MALT lymphoma depends on two main aspects: the primary involved organ and the extension of disease . Gastricof stage . There wof stage , becauseof stage . In the of stage . On the of stage , 10. If In the present case, laparoscopic partial hepatectomy was performed unexpectedly for primary hepatic MALT lymphoma. There are only a few case reports of pure laparoscopic hepatectomy for primary hepatic MALT lymphoma –14 Tabl. Due to Primary hepatic MALT lymphoma is a very rare disease, and it is often difficult to diagnose precisely without biopsy. However, the problem of liver biopsy is dissemination. Laparoscopic hepatectomy has been widespread recently as minimally invasive surgery, so laparoscopic hepatectomy should be considered for the treatment of a resectable liver tumor if there is confusion about the diagnosis.


Dura-attached supratentorial extra-axial ependymoma is a very rare type of tumor, with only nine reported cases. Preoperative diagnosis of dura-attached supratentorial extra-axial ependymoma is difficult and often radiologically misdiagnosed as a meningioma. We report a case of dura-attached supratentorial extra-axial ependymoma that was misdiagnosed using intraoperative histological and cytological examinations.C11orf95-RELA fusion gene. The final diagnosis was RELA fusion-positive ependymoma, World Health Organization grade III.A 26-year-old Japanese man with headache and nausea was referred to our medical facility. Magnetic resonance imaging revealed a cystic mass of 70 × 53 × 57 mm in the left temporoparietal lobe. A peritumoral band with hyperintensity on T2-weighted imaging was observed at the periphery of the lesion, suggesting an extra-axial lesion with no apparent connection to the ventricle. A dural tail sign was also noted on the gadolinium-enhanced T1-weighted image. Preoperative clinical diagnosis was meningioma. Proliferated tumor cells in sheets with intermingled branching vessels were observed in the frozen tissue. Perivascular rosettes were inconspicuous, and the tumor cells had rhabdoid cytoplasm. The tumor was intraoperatively diagnosed as a meningioma, suspected to be a rhabdoid meningioma. Perivascular rosettes were evident in the formalin-fixed paraffin-embedded tissues, suggesting ependymoma. The tumor cells had eosinophilic cytoplasm without a rhabdoid appearance. Anaplastic features, such as high tumor cellularity, increased mitotic activity, microvascular proliferation, and necrosis, were observed. Ependymal differentiation was confirmed on the basis of ultrastructural analysis. Molecular analysis detected Owing to its unusual location, dura-attached supratentorial extra-axial ependymomas are frequently misdiagnosed as meningiomas. Neuropathologists should take great precaution in intraoperatively diagnosing this rare subtype of ependymoma to avoid misdiagnosis of the lesion as other common dura-attached tumors. Ependymoma is a glial tumor in the central nervous system and corresponds to World Health Organization (WHO) grade II category . AnaplasRELA fusion gene [RELA fusion-positive ependymomas have become a distinct entity in the classification of ependymal tumors since 2006 [RELA fusion gene detection [Molecular classification of tumor has been proposed as a prognostic tool for ependymal tumors . Poor prion gene . RELA funce 2006 , and areetection .Ependymomas usually arise in or near the ventricular system . IntracrC11orf95-RELA fusion gene.Here, we present a case of dura-attached SEAE that was initially misdiagnosed as meningioma on the basis of intraoperative histological and cytological findings. Postoperative evaluation of the paraffin-embedded specimens confirmed this case as an anaplastic ependymoma with dural invasion. Ultrastructural analysis supported the ependymal differentiation of tumor cells, and molecular assays detected A 26-year-old Japanese male presented with severe headache and nausea for approximately 3 weeks. He had a prior episode of suspected convulsion of which magnetic resonance imaging (MRI) revealed a cystic nodular lesion measuring approximately 60 mm in diameter, located in the left parietal lobe of the cerebrum. The patient was administered medication, and symptoms were subsequently relieved. However, 12 days later, he experienced worsened symptoms and was transferred and admitted to the emergency department of the University of Miyazaki Hospital.MRI showed a mass of 70 × 53 × 57 mm in the left temporoparietal lobe Fig. a–c. A soIntraoperative histological specimens were immersed in optimal cutting temperature compound , fixed in liquid nitrogen, and cryosectioned. Intraoperative cytological specimens were either squashed or imprinted and immediately fixed in 95% ethanol prior to hematoxylin and eosin (HE) staining.Histological findings of the frozen tumor tissue revealed sheet-like proliferation of tumor cells with branching blood vessels Fig. d. PerivaIn FFPE tissues, high cellularity was observed, with evidence of tumor cell proliferation and branching vessels Fig. a. PerivaImmunohistochemistry was performed as previously reported . The tumA portion of the intraoperative specimen kept at 4 °C was used for ultrastructural analysis . The tumRELA fusion gene, DNA was extracted from five FFPE sections of 5 μm thickness using a blackPREP FFPE DNA kit  [C11orf95-RELA fusion gene was detected  . Polymerted Fig. a, b.Fig.RELA fusion-positive ependymoma, WHO grade III.The final pathological diagnosis was et al. [We report a case of intraoperatively misdiagnosed rare dura-attached SEAE. The tumor was confirmed as anaplastic ependymoma with dural invasion on the basis of the evaluation of the postoperative histological diagnosis of FFPE specimens. A similar case was reported by Salunke et al.  as the iAn accurate intraoperative diagnosis for dura-attached SEAEs is often challenging, owing to bias by the preoperative information, insufficient time, and lacking ancillary techniques. In our case, to minimize the effect of freezing/drying artifacts during intraoperative diagnosis , tissue RELA fusion-positive ependymoma is considered the worst prognosis category of the three molecular subgroups of supratentorial ependymoma [RELA fusion-positive ependymoma and who had undergone with gross total resection [C11orf95-RELA fusion gene was previously reported in one SEAE case with a history of 6-month follow-up and no recurrence [RELA fusion gene as a prognostic factor remains ambiguous in supratentorial ependymoma.endymoma . Howeveresection , 18. C11currence . The preWe report a case of dura-attached SEAE misdiagnosed as meningioma on the basis of intraoperative diagnosis using frozen tissue sections and cytology. Neuropathologists should take great precaution in intraoperatively diagnosing this rare subtype of ependymoma to avoid misdiagnosis of the lesion as other common dura-attached tumors.


Extraneural metastasis (ENM) of glioblastoma are rare. However, as patient overall survival improves, the incidence of ENM has gradually increased. Although several risk factors have been proposed, venous sinus invasion was regarded as a very exceptional route for ENM.We report a 60-year-old man with glioblastoma in the temporal lobe, invading the transverse and sigmoid venous sinus. After gross total tumor resection, the patient received the standard chemoradiation therapy. Systemic evaluation for persistent shoulder and back pain revealed widespread metastasis to lymph nodes and multiple bones 9 months after surgery. Despite spine radiation therapy, the patient became paraplegic and died 1 year after surgery.Venous sinus invasion should be kept in mind by physicians, as a risk factor for glioblastoma ENM. Systemic evaluation of these patients with extracranial symptoms should be performed without hesitation. Glioblastoma is the most common and fatal primary central nervous system (CNS) cancer with highly infiltrative growth. After maximal safe resection, concurrent chemoradiotherapy and adjuvant chemotherapy using temozolomide (TMZ) has been used as standard treatment for initially diagnosed glioblastoma. Despite the improvement of surgical intervention and various treatment strategies, the average survival period is around 15-18 months and 5-year survival rate was reported as 6.8% in the developed country  of glioblastoma was rare  possibly due to strong barriers formed by the dura mater and blood-brain barrier (BBB) , 2. HoweA 60-year-old man presented with a 2-month history of cognitive impairment. He had hypertension and a history of early gastric cancer surgery 10 years previously. There were no abnormal radiological findings on abdominopelvic or chest computed tomography (CT). Brain magnetic resonance imaging (MRI) showed a large contrast-enhancing (67×48×60 mm) lesion in the left temporal lobe, invading the transverse and sigmoid venous sinuses -wildtype glioblastoma with O6-methylguanine-DNA methyltransferase (MGMT) promoter methylation. Tumor cell invasion of the dura was confirmed histopathologically -CT, and chest and abdomen CT were done 9 months after surgery and revealed widespread metastases to the chest & cervical lymph nodes and multiple bones, including the vertebrae, scapula, and pelvis Figure 3Previous studies reported that risk factors for glioblastoma ENM include invasive procedures, RT, young age, prolonged survival time, tumor recurrence, and sarcomatous component , 9. AlsoGlioblastoma ENM probably have a histopathological and/or genetic predisposition. To develop distant metastases in systemic cancers, epithelial-mesenchymal transition (EMT) can be required. Sarcomatous metaplasia in glioblastoma is an example of EMT. With a sarcomatous phenotype, glioblastoma cells can degrade extracellular matrix proteins, promoting vascular or lymphatic invasion. Some tumor clones have some specific genotypes that predisposes to ENM. In analysis of genetic predisposition, glioblastoma with ENM had a tendency of tumor suppressor gene alteration. Common genomic alteration included TP53, ATRX, PTEN, RB1, TERT, IDH1, and NF1 . Among tvia a vascular or lymphatic pathway  and wide dissemination feature in ENM, the metastatic route is probably  pathway . In this pathway , previou pathway Table 1. pathway . In a tuThe prognosis of glioblastoma with ENM is dismal. Median overall survival from diagnosis of ENM ranged from 1.5 months to 6 months. Although there was no prognostic significance in age, gender, initial tumor location, or time interval from initial diagnosis of primary tumor to detection of ENM, the presence of sarcomatous component and lung metastasis were possible poor prognostic factors , 13, 21.Venous sinus invasion should be considered as a possible route for ENM in glioblastoma. In managing patients with such risk factor, clinicians should not hesitate to perform a systemic evaluation when patients have extraneural symptoms.The raw data supporting the conclusions of this article will be made available by the authors, without undue reservation.Ethical review and approval were waived for this study, due to the fact that this is a single case report. Written informed consent was also obtained from the participant for the publication of this manuscript, including any potentially-identifying images or data. The patients/participants provided their written informed consent to participate in this study. Written informed consent was obtained from the individual(s) for the publication of any potentially identifiable images or data included in this article.Conceptualization: K-HL & K-SM. Data curation: KA, YK, and K-SM. Analysis and interpretation of data: K-HL & K-SM. Funding acquisition: K-SM. Writing-original draft: KA, YK, and K-SM. Writing-review & editing: K-HL. Manuscript approval: all authors. All authors contributed to the article and approved the submitted version.This work was supported by Basic Science Research Program through the National Research Foundation of Korea (NRF) funded by the Minist . There was no role of the funding bodies in the design of the study, in the collection, analysis, and interpretation of data, or in the writing of the manuscript.http://www.textcheck.com/certificate/qoCxX6).The English in this document has been checked by at least two professional editors, both native speakers of English. For a certificate, please see: (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.


Multifocal desmoplastic infantile ganglioglioma/astrocytoma (DIA/DIG) has rarely been reported. Here, two cases have been presented, reviewing the literature and proposed treatment algorithms for this rare tumor.We report two patients diagnosed with multifocal DIA/DIGs in West China Hospital. In addition, a literature review was performed, in October 2019, on case reports of DIA/DIGs with multifocal lesions. The clinical and radiological features, treatment, and outcome of this rare disease were discussed.DIA/DIGs with multifocal locations were rare, and only thirteen cases (including ours) had been reported. This series included 8 males and 5 females with a mean age of 31.4 ± 45.7 months . The supratentorial hemisphere, suprasellar region, posterior cranial fossa, and spinal cord were frequently involved. Ten patients (76.9%) received surgical resection for the symptomatic lesions and three patients (23.1%) underwent biopsy. Seven patients received chemotherapy postoperatively. Six individuals had tumor recurrences during the follow-up period, while three patients had tumors that spontaneously regressed. Finally, two patients died of tumor progression and one patient died of respiratory insufficiency and hypothalamic dysfunction.Multifocal DIA/DIGs have more aggressive clinical behavior and poor outcome despite benign histology. DIA/DIGs should be included in the differential diagnosis of multifocal brain tumors in children. The mainstay of treatment is surgical resection; adjuvant treatment with chemotherapeutic drugs is unknown and requires additional research. Desmoplastic infantile astrocytoma (DIA) and desmoplastic infantile ganglioglioma (DIG) are rare intracranial tumors in infants, and account for only 1.25% of all childhood brain tumors . They frOnly 11 cases of DIA/DIGs with multifocal intracranial or intraspinal tumors have been recorded in the English literature –11. Its . Postoperative histopathological examination supported the diagnosis of DIA/DIG . The patient was discharged from the hospital with a favorable outcome two weeks later and a close follow-up was achieved.A 12-year-old female was admitted to our hospital with seizures over two months. Cranial magnetic resonance imaging (MRI) depicted five solid tumors in the right and left tentorium, left para sellar, right lateral ventricle, and left frontal lobe respectively, the solid tumors are hypointense in T1 sequences and isodense or hypointense with obvious peritumoral edema, after administration of gadolinium, the tumors enhanced strongly (. Postope). The suprasellar region tumors contained both solid and cystic components, the solid part showed hypointense in both T1 and T2 sequences, the cystic part showed hypointense on T1 sequences and hyperintense in T2. CT angiography depicted no obvious vessels inside the tumors  guidelines . The litCase report or article titles and abstracts were evaluated. Only articles are written in English that described cases of multifocal desmoplastic infantile ganglioglioma/astrocytoma were included. The literature review was conducted independently by two authors  using EndNote X7 software . For controversial studies regarding the inclusion, a discussion would be performed until a consensus agreement was reached. The patient characteristics such as demographic data, clinical and radiological features, pathologic diagnosis, treatment and prognosis were extracted from the literature by two authors  independently.A total of 13 cases (including ours) Ten patients 76.9%) in this series received surgical resection for the symptomatic lesions and three patients (23.1%) underwent biopsy. Case 1 has been previously reported by our group member , but we % in thisBased on the postoperative histological investigation, all of the patients were categorized as WHO grade I DIA/DIGs. Histologically, they frequently showed a desmoplastic spindle cell tumor with a densely eosinophilic collagenous substrate  reportedTaratuto et al. reported the first case of desmoplastic infantile astrocytoma (DIA) in 1984  and VandDIA/DIGs frequently occur as a supratentorial, contrast-enhanced solid tumor with cystic components . And theOur study found no obvious differences regarding histological features between multifocal DIA/DIGs and solitary counterparts. Both of them contained reticulin- and collagen-rich areas with spindle fibroblastic elements, DIGs always show neuronal elements whereas DIAs show astrocytic component . Besidesvia the CSF pathway, and they identified tumor cells in the CSF of these patients , the Medical Research project of Sichuan Province (Fund No. Q20042), the Science and technology Project of Tibet Autonomous Region. The central government guides local projects (Fund No. XZ202102YD0032C).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.


Advantages of three-dimensional (3D) computed tomography-based navigation system has recently been used for safe, accurate, reliable spinal tumour excision. This study presents two cases, a 33-year-old male and a 17-year-old male presented in the clinic with mid-back and low back ache, respectively with change in posture. Radiology suggested an osteoid osteoma. Accurate localisation and complete extirpation of the lesion were performed using a translaminar approach with O-arm Navigation. After follow-up of one year, both did not complain of back pain or radiation, scoliosis had improved in both cases and their VAS was 0 and ODI was 0%. 3D navigation with the O-arm system provided an easy and accurate localisation of the lesion, reducing the risk of instability subsequently and avoiding instrumented stabilisation. This technique also provided for histopathological confirmation of the diagnosis. Over the decades, surgical techniques of the spine have evolved, and minimally invasive procedures are now utilised for excision of osteoid osteoma with high precision. We report two cases of osteoid osteoma of spine managed with surgical excision with O-arm guided navigation. Case 1 had osteoid osteoma at superior articular process of the D11 vertebra while Case 2 had osteoid osteoma of the L4 right pars interarticularis, both treated with surgical excision with O-arm guided navigation.Osteoid osteoma is a primary benign bone lesion that commonly occur in the second decade of life, but also varies from the first to fourth decade with a male to female predominance of 2:1. The clinical symptom reported is night pain, caused by nidus induced prostaglandin/prostacyclin production. Patients also present with painful scoliosis due to soft tissue and muscle irritation, leading to asymmetrical spasm. Surgical excision is the definitive method of treatment for patients unresponsive to nonsteroidal anti-inflammatory drugs like aspirinA 33-year-old male presented with dull mid-back pain radiating to the left side of the chest for 7 months. Pain was aggravated at night and relieved by taking nonsteroidal anti-inflammatory drugs (NSAIDS). Pain progressively increased to the point of causing difficulty in daily activities. On clinical examination, he had dorsolumbar scoliosis with convexity to the right, tenderness, and paraspinal spasm in that region. The scoliosis was partially corrected on bending forward. His back visual analog scale (VAS) score was 8 with Oswestry Disability Index 40%. Neurological examination was normal. Radiographs of the dorsolumbar spine suggested sclerosis in the D10 and D11 facet joint on the left side with scoliosis with convexity to the right. Pre- and post-contrast magnetic resonance imaging of the dorsolumbar spine was performed. The dynamic evaluation was performed with contact using time-intensity curves and correlated with plain CT. There was a well-defined lytic lesion seen involving the superior articular process of the D11 vertebra measuring 11mm x 11mm x 9mm being hypointense on T2 and mildly hyperintense on T1 with central sclerosis on CT scan scalloping the inferior articular process of the D10 vertebra. It showed rapid early uptake of contrast on arterial phase with slow washout. These findings favoured a diagnosis of osteoid osteoma .A 17-year-old male presented with low-back pain for eight months. Pain was aggravated at night and relieved by taking nonsteroidal anti-inflammatory drugs (NSAIDS). Pain progressively increased to the point of causing difficulty in daily activities. Clinical examination showed lumbar scoliosis with convexity to left, tenderness, and paraspinal spasm in that region. The scoliosis was partially corrected on bending forward. His back visual analog scale (VAS) score was 9 with Oswestry Disability Index 44%. Neurological examination was normal. Radiographs of the lumbar spine suggested sclerosis in the L4 right pars interarticularis and lumbar scoliosis with convexity to the left. Pre- and post-contrast magnetic resonance imaging of the lumbar spine was performed. The dynamic evaluation was performed with contact using time-intensity curves and correlated with plain CT. There was an area of well-defined radiolucency with central sclerotic focus seen involving the L4 right pars interarticularis measuring 4mm. It showed rapid early uptake of contrast on arterial phase with slow washout. These findings favoured a diagnosis of osteoid osteoma which was confirmed on histopathology .The patient was positioned prone on the Jackson table . After taking safe surgical precautions, a single two-dimensional fluoroscopy image was taken from the O-arm  to mark the incision and a stab incision was made cranially to attach the reference probe to the spinous process, extending caudally to the dorsolumbar region. Subsequent dissection was performed to reach the left D10 D11 facet joint for first case. For the second case, the Medtronic METRx 14mm tube system was used with Wiltse approach. With O-arm setting changed to minimise the radiation dose; a CT scan was taken within 15s. The navigation system used was StealthStation S8 . The basic data used for navigation consisted of pre-operative CT data, which were transferred and recorded on the system computer and reconstructed into three-dimensional (3D) images.After registering the navigation with a 1.5mm ball-tipped reference probe, the accuracy of the images was verified by point merging and surface merging. The lesion was identified and accurately localised with the probe. Under neurosurgical microscope OPMI Pentero 900  careful drilling was initiated with 3mm diamond tip MIDAS Navidrill  till the lesion wall was reached as identified with the probe. The lesion was extirpated en masse in both cases. Further drilling was done in the walls of the articular process taking care not to breach the pedicle and keeping the dura safe.Adequacy of extirpation was checked using the Navigation probe. Total amount of blood loss was less than 30ml in both cases. The operative time for the first case was 90min and the second case was 75min. Surgical wound was closed. Both patients walked 4h after the surgery. On the second day of surgery, the back VAS improved to 2, and patients were discharged home. Histopathological examination confirmed osteoid osteoma diagnosis. After follow-up of one year, both did not complain of back pain or radiation, scoliosis had improved in both cases VAS was 0 and ODI was 0%2. CT-guided percutaneous resection technique has limitations such as accurate localising the drill tip leading to malposition and inadequate drilling with increased risk of inadvertent dural and spinal cord injury, or increased risk of infection due to substandard sterility precautions in radiological suites. CT-guided percutaneous radiofrequency thermal ablation and laser photocoagulation for the treatment of osteoid osteomas located in the extremities has been used successfully. However, proximity to neural structures makes this modality conflicting in the spine. Thermal damage to the neural structures due to the heating tip has always raised concerns. Inadvertent injury to adjacent neural structures, high failure rates in the spine and lack of histologic confirmation of diagnosis makes it less ideal for treating osteoid osteomas in the spine.Treatment of an osteoid osteoma involves complete intralesional excision of the nidus, either by surgical or radiological intervention after failure of adequate medical management. Although, intra-operative nidus localisation is challenging and can lead to extensive resection of the adjacent normal bone structure leading to risk of iatrogenic fracture and extended period of healing, instability requiring stabilisation, or neurovascular injury. Accuracy of intra-operative localisation of the nidus include radionuclide localisation with a gamma probe, intra-operative CT-guided localisation, or a combination of scintigraphy and computer navigation3. Gasbarrini et al4 studied 81 patients operated for osteoid osteoma either by conventional excision therapy or minimally invasive surgery (six of 81). They concluded that minimally invasive surgery has advantages of reducing soft tissue morbidity and the collateral damage caused by conventional surgical approach, it allows patients a more rapid and complete return to normal function. Considering accuracy of 3D fluoroscopic navigation, Mason et al5 analysed 30 studies for accuracy of pedicle screws. They reported accuracy of conventional fluoroscopy as 68.1%; accuracy of 2D fluoroscopic navigation as 84.3%; and with 3D fluoroscopic navigation accuracy as 95.5%. They concluded that accuracy rates of 3D as compared with 2D fluoroscopic navigation were consistently higher throughout all individual spinal levels. This result show that 3D fluoroscopic navigation can help surgical excision of osteoid osteoma more accurately.Advantages of a three-dimensional (3D) computed tomography-based navigation system has recently been used for safe, accurate, reliable spinal tumour excisionLimitations of this study includes lack of long-term follow-up to evaluate for recurrence. Also, large group of patients is required to make this a standardised treatment for osteoid osteoma.This study of 3D navigation with the O-arm system provided precise, safe, accurate, reliable localisation of the lesion, reducing approach related morbidity, the risk of iatrogenic instability and subsequent instrumented stabilisation, thereby reducing the hospital stay and cost of treatment. An intra-operative verification O-arm spin confirmed complete tumour excision and a low dose protocol of the O-arm significantly reduced the radiation exposure to the patient. Histopathological confirmation of the diagnosis can also be provided by this technique.


Enlarged cisterna chyli is an infrequently encountered entity and is most often an asymptomatic, incidental finding on imaging for other reasons. The pathogenesis of cisterna chyli enlargement is not well elucidated and includes infectious, inflammatory, and idiopathic causes. In this report, we present the rare case of an asymptomatic, markedly dilated “mega” cisterna chyli in a 60-year-old female. Enlargement of the cisterna chyli (CC) is a rare condition and is most frequently encountered as an incidental finding on cross-sectional imaging performed for other reasons. The CC is a saccular dilatation of a lymphatic duct that receives lymphatic flow from lumbar and intestinal lymphatic trunks and serves as the origin of the thoracic duct . The etiThe patient is a 60-year-old Caucasian female with a history of hypertension, hyperlipidemia, and medically-managed Crohn’s disease who self-referred to our institution for evaluation of an incidentally encountered 12.8 cm esophageal duplication cyst. This was first discovered on a routine abdominal computerized tomography (CT) scan five years prior to her presentation. She had undergone two prior endoscopic ultrasound (EUS)-guided fine-needle aspirations (FNA) of this lesion. During the initial endoscopy, a cystic lesion under the diaphragm was identified, measuring 6 x 3 cm, thought to be a duplication cyst. EUS-FNA yielded clear, watery fluid with fluid analysis and triglyceride levels of 67 mg/dL. The cyst recurred and she underwent repeat EUS-FNA, and fluid analysis at that time demonstrated an elevated triglyceride level, 173 mg/dL. The patient denied any current symptoms including dysphagia, weight loss, or chest pain. There were no abnormal findings documented on physical examination, including no evidence of abdominal distension, ascites, or lymphedema. A presumptive diagnosis of recurrent enlarged CC was made, and the patient underwent abdominal magnetic resonance imaging (MRI) which demonstrated a large tubular cystic structure extending from the inferior mediastinum between the aorta and inferior vena cava terminating behind the crus of the diaphragm at the level of the T12/L1 vertebrae. This lesion measured 12.8 cm craniocaudally, 3.4 cm transversely, and 2.7 cm anteroposteriorly  in a 60-year-old patient. We utilized an indexed Pubmed search for keywords pertaining to the anatomy and physiology of the CC, the pathophysiology of enlarged CC, and treatment options for symptomatic disease. As no private health information was documented for this case report, informed consent was waived. The CC is classically described as a saccular dilatation found at the origin of the thoracic duct (TD) in the retrocrural space, most frequently found at the T11-L2 vertebral levels , 6.Enlargement of the CC is a rarely documented occurrence, despite its heterogeneous morphology. An investigation of the CC in 40 patients through multidetector row CT (MDCT) imaging demonstrated maximal mean maximum dimensions of 3.8 mm AP x 4.0 mm TD x 14.1 mm in length (ranging from 1.7 x 7.8 x 1.3 mm to 6.9 x 3.8 x 30.1 mm) . To our The etiology of enlarged CC is poorly understood and is often encountered incidentally in the diagnostic workup for other pathologies. Impaired lymphatic flow, intrathoracic lymphatic obstruction, prior radiation/trauma/injury to the thoracic duct, or increased lymph production above the capacity of the CC may be implicated. We suggest the following framework for understanding the pathogenesis of enlarged CC: (1) intrinsic: idiopathic or known weakness in lymphatic duct wall integrity or (2) extrinsic: dilatation due to increased lymph production or impaired outflow due to distal obstruction or injury of the thoracic duct or increased central venous pressure. In our case, there was no clear evidence that the patient had any intrinsic lymphangiopathy, thoracic duct pathology resulting in distal obstruction, or increased production of abdominal or lumbar lymph fluid.Intrinsic causes of enlarged CC may be both a reflection of physiologic variations or primary lymphangiopathy. During embryogenesis, major lymphatic sacs are segmented by connective tissue to form lymph node stations, apart from the superior portion of the CC . AnatomiExtrinsic causes of enlarged CC include increased lymph production beyond the capacity of the sac resulting in dilatation. Theoretically, any increased lymphatic production in this regard could precipitate enlargement of the CC. A documented example of this is cirrhosis. Verma et al. documented that cirrhosis was associated with CC dilation greater than 2 mm and this dilation was significantly more frequently encountered in uncompensated cirrhotic patients compared to those with compensated cirrhosis  . A putatSymptomatically enlarged CC are rare, and most lesions are found incidentally on imaging for other indications. Rarely, enlarged CC can result in pain, gastrointestinal discomfort, nausea, or weight loss , 17. In The diagnosis of enlarged CC should be made at the exclusion of other pathologies. Key imaging characteristics may aid in narrowing the differential diagnoses. These include retrocrural lymphadenopathy in the setting of malignancy or infection ,8. The iThe normal appearance of the CC on routine MRI of the abdomen has also been well-described . In thisThere are multiple features in this case that suggested a benign etiology; the lesion was rounded with a smooth contour and well-defined margins, with no evidence of invasion of adjacent mediastinal or retrocrural structures, exhibited no internal enhancement, and was of homogenous fluid attenuation Figure . This leAs the vast majority of enlarged CCs are asymptomatic, treatment is seldom required and infrequently reported in the literature. Rare complications including persistent chylothorax, chylous ascites, or gastrointestinal symptoms may necessitate intervention. Conservative approaches include chest tube drainage, medium-chain fatty acid diet, parenteral nutrition, and somatostatin analogue therapy (octreotide) . No repoWhile transthoracic surgical ligation of the TD is well-established, surgical ligation of the CC is rarely performed and reserved for chylothorax or ascites refractory to conservative or radiographic management, often in pediatric cases , 24. ZanIn addition to upfront ligation, Ishiura et al.  reportedWe report here the rare case of a 60-year-old female incidentally found to have an extremely enlarged (12.8 x 3.4 x 2.7 cm) "mega" cisterna chyli, manifesting as a recurrence after prior endoscopic drainage. Enlargement of the cisterna chyli is extremely uncommon. While most cases are idiopathic, intrinsic and extrinsic factors may predispose CC dilatation. Most enlarged CCs can be monitored. In rare symptomatic cases of enlargement  or in the setting of iatrogenic injury or pathologic obstruction, conservative management consists of conversion to medium chain fatty-acid diet, parenteral nutrition, chest tube drainage, and somatostatin analogue therapy. Refractory cases may be amenable to radiographic embolization. Rarely, surgical ligation of the CC, through open or laparoscopic approaches, or lymphovenous bypass may be necessary.


We report a case of a jejunal hematoma in a six-year-old boy with an antecedent history of trauma. The development of duodenal hematoma post blunt abdominal trauma is well known; however, the jejunal hematoma is very rare. A six-year-old boy was brought to Tata Main Hospital (TMH), Jamshedpur, with pain abdomen, associated with a history of trauma to the abdomen. There were no signs of peritonitis on clinical examination. Initial ultrasonology revealed mild free fluid in the abdomen. CT abdomen was suggestive of intramural hematoma in the jejunum. Exploratory laparotomy findings were in concordance with CT abdomen findings. Resection-anastomosis of the jejunum was done. The patient was discharged uneventfully on postoperative day 7.Blunt trauma to the abdomen is the principal cause of jejunal hematoma. The trauma in the majority of cases is trivial and usually the patients present late. The symptoms range from mild abdominal pain to intestinal obstruction with acute abdomen. A trial of conservative management is justifiable in a stable patient. If no clinical improvement is seen, surgical intervention is indicated. Surgical exploration was necessary because of signs and symptoms of intestinal obstruction due to jejunal hematoma. In pediatric cases, loss of functional bowel length and sudden decompensation due to expanding hematoma favor early exploration. Intra-abdominal bowel and solid organ injuries after blunt abdominal trauma are significant causes of mortality/morbidity in the pediatric age group. Hematoma is predominantly seen in solid intra-abdominal organs and fixed and relatively immobile segments of the gut, like the duodenum . PerforaA six-year-old boy presented to us with a complaint of pain abdomen for seven days. There was a history of injury from the bicycle handlebar about 10 days ago. Initially, the boy was managed with enteral analgesics at a local hospital on an outpatient basis. The boy was tolerating oral feeds, with normal bowel and bladder movements. However, the boy was referred to us due to persistent pain.Apart from the mild pallor, the general clinical examination was unremarkable with stable vital parameters. The abdominal examination was normal and there was no evidence of focal tenderness or lump. Ultrasonography abdomen was suggestive of mild free fluid. Hemoglobin level at presentation was 10.5 g/dl, total leucocyte counts 9,200/cmm, with 73% neutrophils. Platelets were adequate with counts of 185,000/cmm. INR was 1.1, while APTT was 27s .On day 1 of admission, the boy had an episode of bilious vomiting. X-ray abdomen showed multiple air-fluid levels Figure , 3. The Blunt injuries to the abdomen are a significant cause of morbidity and mortality in children. The most common mode of presentation is perforation in hollow viscus and laceration, hematoma in solid organs. There may be a significant delay between the onset of injury and the onset of symptoms as most abdominal trauma may be mild or trivial, that go unnoticed by the patient or the family. In our case, the boy presented to us after 10 days of abdominal trauma with a handlebar. Although he continued to experience pain abdomen, it did not affect his daily activities. The history of trauma with a handlebar could be elicited only after detailed questioning as the parents assumed that the trauma due to the handlebar was inconsequential.In a solid organ injury following blunt abdominal trauma, laceration and hematoma are usually seen while hollow viscus presents with perforation. Hematoma is frequently seen in the retroperitoneal segment of the duodenum. Bleeding diathesis needs to be ruled out in children presenting with hematoma . In contSeemingly inconsequential blunt abdominal trauma may lead to intramural jejunal hematoma in children, and it may result in intestinal obstruction after some time lag between injury and presentation. Surgical intervention is the preferred approach in a child who develops intestinal obstruction; however, there is a risk of loss of functional bowel segment and decompensation due to expanding hematoma in the conservative approach.


Sex cord-stromal tumors are always found in ovary, but the occurrence of this kind of tumor at extraovarian locations is extremely rare. Up to now, the case concerning fibrothecoma of broad ligament with minor sex cord elements has not been reported, and it is extremely challenging to diagnose before surgery. In this case report, we summarized pathogenesis, clinical features, laboratory finding, imaging studies, pathology, and therapeutic schedule of this tumor, with the aim of raising awareness and attention to this type of disease.A 45-year-old Chinese woman was referred to our department with intermittent lower abdominal pain for about 6 years. On examination, both ultrasonography and computed tomography revealed she had a right adnexal mass.Based on the results of histology and immunohistochemistry, the final diagnosis was confirmed as fibrothecoma of broad ligament with minor sex cord elements.This patient underwent laparoscopic unilateral salpingo-oophorectomy with excision of the neoplasm.Eleven days post-treatment, the patient complained that the symptoms of abdominal pain was disappeared. There is no evidence of disease recurrence 5 years after laparoscopic surgery according to the consequences of radiologic examinationThe natural history of this kind of tumor is uncertain. Although main treatment of this neoplasm might be surgical resection and good prognosis can be achieved, we believe that long-time follow-up is extremely important in all patients diagnosed as fibrothecoma of broad ligament with minor sex cord. Laparoscopic unilateral salpingo-oophorectomy with excision of the tumor should be recommended to these patients. The morphology of fibromas and thecomas is similar, which give rise to the use of the jargon fibrothecoma. This type of tumor represents approximately 3% to 4% of all ovarian lesions. Another subgroup is defined as stromal tumor with minor sex cord elements, which is made up of fibrothecomatous and/or fibroma with minor sex cord elements (<10%). However, sex cord-stromal tumors are exceedingly rare in an extraovarian location. To the best of our knowledge, only 4 cases of stromal tumors in the broad ligament (3 cases are thecoma and 1 case is fibrothecoma) have been documented,–10 but no case of fibrothecoma of broad ligament with minor sex cord elements has been reported in the published literature. Here, we will discuss this kind of tumor and review the literature.Ovarian sex cord-stromal tumors, which can present as various morphology and clinical behavior, account for 7% of all ovarian tumors.A 45-year-old Chinese woman with unremarkable past medical history, gravida 4 para 2, was admitted to our department with intermittent lower abdominal pain for about 6 years and recent (2 months) progressive deterioration. Visual inspection showed that cervix was slightly enlarged accompanied with erosion. During bimanual examination, a 3 cm diameter, solid and non-mobile tumor could be palpated at the right lower quadrant. Peripheral blood cell counts showed hemoglobin was 97 g/L, however, white cell count and platelet cell count was normal, as tumor makers, including carbohydrate antigen 125, carcinoembryonic antigen, and α-fetoprotein, were within normal range.Ultrasound imaging demonstrated there was a round mass (about 37 × 36 mm) in the right adnexa and the lesion was avascular. The plain computed tomography (CT) scan also revealed a low density neoplasm with well-demarcated margin, sized 3.7 × 3.0 cm, in the same location, suggestive a broad ligament leiomyoma Fig. A. ContraDuring intraoperative period, local adhesions could be observed between the right adnexa and the bowel and mesentery. After carefully separating the adhesions step by step, we observed that there was a tumor, sized 4 × 4 × 3 cm, with a clear margin, on the right broad ligament, but the fallopian tubes, ovaries and uterus were normal and not invaded Fig. A. The exThe resected specimen consisted of the lesion and the right adnexa, and the tumor had no capsule. Microscopic examination showed that it was a mesenchymal neoplasm mainly made up of epithelioid cells Fig. A, which The diagnosis of fibrothecoma of broad ligament with minor sex cord elements was made in our patient on the grounds of expression of several markers that are the characteristics of ovarian sex cord-stromal tumors and ruling out of other neoplasms.Six days after the operation, the patient complained that the symptoms of abdominal pain was relieved. Eleven days post-treatment, the abdominal pain was disappeared. After she was discharged from the hospital, she had ultrasonography or CT examination every 3 months. There is no evidence of disease recurrence 5 years after surgery.,9 and another case was reported in Italian. Two were classified as fibrothecoma, two as thecoma,,10 but our case was diagnosed as fibrothecoma of broad ligament with minor sex cord elements. There was a large mass in 1 patient, and the symptoms in the other 3 patients were not clear due to the unavailable literature , but our patient had a chief complaint of abdominal pain. The sizes were different between these tumors, and the diameter of one of the case was 20 cm, which is the 5 times the diameter of our case. Patients with no obvious symptoms or presenting as slight symptom may be the reasons why the neoplasms could be large in size.Primary tumors that occur in broad ligament are extremely rare, which include leiomyoma, cyst, neurilemmoma, teratoma, lipoma and hemangioma and so on, but before now, only 3 cases of stromal tumors in the broad ligament have been documented in English literature, However, if a lesion is considered as primary broad ligament tumor, the most important thing is that the ipsilateral ovary should appear normal, with wholly intact cortex. The histogenesis of extraovarian sex cord-stromal tumors is still uncertain, but ectopic gonadal stromal tissue is thought to be one of the histogenetic origin of these tumors.,13 Up to now, pelvis and retroperitoneum are the most common location of the origin of extraovarian sex cord-stromal tumors.The broad ligament consisting of 2 segments, one of which is mainly made up of fibroconnective tissue, and another segment contains a large number of vessels, muscles and connective tissue. Extraovarian sex cord-stromal tumors, according to the related references, are exceedingly rare. Meanwhile, different anatomical sites of this kind of tumor have also been reported, which include fallopian tube, mesovarium, retroperitoneum, adrenal gland, mesentery, broad ligament and an umbilical hernia sac.,9 Before operation, the examination of testosterone and estradiol levels had not been performed in our patient, but both of them were within the normal range when they were tested postoperatively, which indicated that our case did not generate hormone.A review of the cases showed that laboratory results of serological tumor markers, which include carcinoembryonic antigen, α-fetoprotein and carbohydrate antigen 125, were normal. Two of the cases (fibrothecoma) had no function of hormone production, and 1 case (thecoma) was found to produce estradiol and estrone. if a lesion without calcification could cast a dense acoustic shadow in ultrasonography, it could be regarded as a rather specific finding of the ovarian thecoma. CT might be the optimal diagnostic option for the diseases, and CT was carried out in one of the case according to the literature. Generally, CT appearances of pure thecomas or thecomas with scarce fibrotic components of ovary are not distinct, and precontrast CT of our patient revealed a low density neoplasm with well-demarcated margin, as the lesion was slightly enhanced on the arterial phase as well as the venous phase. A research by Zhang et al prospectively estimated the magnetic resonance imaging (MRI) characteristics of 18 cases of thecomas/fibrothecomas of ovary. The authors drawn a conclusion that 61.1% of thecomas/fibrothecomas were homogenous tumors and appeared isointense to the myometrium on diffusion-weighted imaging MRI. Pure thecomas of ovary tend to demonstrate greater hyperintensity on T2-weighted images. Due to the vascularization of theca cells, a more apparent contrast-enhancement could be seen in thecomas/fibrothecomas. We also think that MRI might exert an pivotal role in the diagnosis of this disease.For sex cord-stromal tumors, it is difficult to differentiate a subtype only in clinical manifestations and laboratory examination, so the imaging findings should be performed to provide valuable diagnostic information. Ultrasound is the most common methods for this type of tumor. According to the literature, ultrasonography was implemented in one of the cases of stromal tumors in the broad ligament. Ultrasonography revealed a big mass and ascites, and most of the neoplasm was solid and partly cystic. In our case, the lesion appears solid and avascular, with a heterogeneous ultrasonic echo. To get a better understanding of the disease, we believe that the enhanced ultrasonography should be recommended. Reviewing the literature, But for extraovarian sex cord-stromal tumors, 1 case (thecoma of broad ligament) showed that the lesion presented as solid nodular parts with brownish-yellow appearance, and yellowish clear fluid was seen in several cystic spaces of the neoplasm. The surface of the neoplasm was white to gray in our case. Pathologically, according to the literature, ovarian sex cord-stromal tumors are made up of uniform spindle cells that can arrange in sheets, and the amount of eosinophilic cytoplasm was scant to moderate, with round nuclei but without nuclear atypia or nuclear grooves. Theca cells that usually surround the ovarian follicles compose ovarian thecomas, which demonstrate estrogenic activity in most cases.,18 On immunohistochemistry, ovarian thecomas could show positivity for vimentin, WT1 and inhibin in. Interestingly, immunohistochemistry in our case reveals positive staining with WT1, which is rarely expressed in sex cord-stromal tumors of broad ligament. Minor sex cord element elements (granulosa cells) have also been seen in our patient.Upon gross examination, ovarian sex cord-stromal tumors are mostly solid masses and the cut surface of them shows grayish white and yellowish areas.Differential diagnoses of the tumor that we report may depend on morphology of the neoplasm and still on the age of different patients. The characteristics of kinds of differential diagnoses are too more to be discussed detailed, and the differential diagnoses probably include epithelioid smooth muscle tumors, malignant melanoma, metastatic carcinoma from other places, a variety of sarcomas, intra-abdominal desmoplastic small round cell tumor, multifarious small round blue cell tumors, malignant mesothelioma, malignant gastrointestinal stromal tumors, small cell carcinoma of the ovary of hypercalcemic type (this is easily to be ruled out if there is no mass on the ovary), and granulosa cell tumors.,20 At the same time, these tumors are more likely to be found in postmenopausal women and most cases are regarded as benign neoplasms. Although only several cases of thecoma/fibrothecoma of broad ligament had been reported, uncertain malignant potential of these neoplasms could not be ignored. Surgery was performed in all of the 3 cases, and in our opinion, surgical resection may be the gold standard for the treatment of these tumors. For extraovarian sex cord-stromal tumors, fertility-sparing surgery, such as preservation of the ipsilateral ovary, is a significant consideration in young patients who desire future childbearing.Given all factors above, it is challenging for gynecologist to make a correct diagnosis on this kind of tumor before surgery or without a biopsy. Thecomas represent approximately 0.5% to 1% of all primary ovarian tumors.Extraovarian sex cord-stromal tumors are rarely documented, we are the first one to report fibrothecoma of broad ligament with minor sex cord elements in the world. No specific clinical symptoms could be observed before surgery. Thus, it is difficult for doctors to make a correct preoperative diagnosis. According to the literature, we believe that CT, MRI and enhanced ultrasonography should be recommended to this type of tumor, especially MRI. Additionally, the final diagnosis must depend on the immunohistochemical staining results. Owing to the semi malignant potential of these tumors, surgery should be performed, and laparoscopic unilateral salpingo-oophorectomy with excision of the neoplasm should be recommended to the postmenopausal women. After operation, long-time follow-up is extremely important in all patients with sex cord-stromal tumors of broad ligament.Pathology department of the second Xiangya Hospital provided the Histological and immunohistochemical figures for the authors.Conceptualization: Jianlin Chen.Data curation: Peng Zhou, Jianlin Chen.Investigation: Peng Zhou.Project administration: Yanjun Chen.Software: Yanjun Chen.Writing – original draft: Yanjun Chen, Jianlin Chen.Writing – review & editing: Yanjun Chen, Jianlin Chen.


Ovarian cysts are common during pregnancy as an outcome of routine prenatal ultrasounds. Although most cases are benign, complications, such as torsion, rupture, and malignant changes, can occur. Torsion risk increases fivefold during pregnancy. It is extremely hazardous to expectant mothers and unborn children. In a rural health tertiary center, we report the case of a 23-year-old primigravida with 14 weeks of pregnancy presented with acute abdomen and nausea, vomiting for four hours. On ultrasonography, she was diagnosed with a 14 cm × 11 cm left dermoid cyst. She underwent a laprotomy, and a twisted dermoid cyst was found; therefore, a left oophorectomy was performed with consent. Histopathological examination revealed the presence of a dermoid cyst. She is regularly followed up at our center with a healthy intrauterine fetus growing within. Although antepartum surgical intervention has been proven safe, there are some risks associated with abdominal surgery for both pregnant women and their unborn children. As a result, the management strategy must be chosen based on a risk-benefit analysis of adnexal mass characterization and gestational age. Dermoid cysts, or mature cystic teratomas, are the most common ovarian tumors among women of reproductive age, making up a remarkable 20% of all adult ovarian tumors ,2. TheseAdnexal torsion around the vascular axis or pedicle is referred to as ovarian torsion. Common risk factors include a free mobile cyst with a long pedicle. Acute abdominal pain during pregnancy can exhibit nonspecific signs and symptoms related to various diseases. The infundibulopelvic and tubo-ovarian ligaments form a central line axis, and when the adnexa, ovary, or fallopian tube rotate completely around this axis, it sparks an event known as adnexal torsion . This leA 23-year-old primigravida presented to the ANC clinic with severe, non-radiating, unrelieved abdominal pain and four episodes of vomiting for four hours. Her symptoms were severe and unrelieved by medication, and she had no history of vaginal discharge or bleeding. The patient conceived naturally without treatment and provided no information about any illnesses, fevers, urinary problems, diarrhea, or constipation. The patient had one visit at eight weeks of gestation and had no relevant medical or surgical history. On general examination, her blood pressure was 90/60 mmHg, pulse rate was 112/min, afebrile, and her cardiovascular and respiratory systems were normal. A tender adnexal cystic mass was detected on abdominal examination, in addition to a 14-week palpable uterus. These findings were corroborated by vaginal examination.All of her blood and urine test results were within normal limits. Ultrasonography revealed a 14 cm × 11 cm single mass lesion in the left iliac fossa with thin-walled septa and dermoid cyst components. It also showed a single intrauterine live fetus of 14 weeks with a placenta developing in the fundoposterior segment. With proper consent and in view of an acute emergency patient was posted for emergency laparotomy under spinal anesthesia after giving a stat dose of intramuscular synthetic hydroxy progesterone, 500 mg. A right ovarian cyst measuring 14 cm x 10 cm was visualized to be triply rotated around its pedicle, as shown in Figure  postoperative day followed by oral tocolytics 10 mg twice a day for 14 days. A normal postoperative obstetric ultrasound suggesting live 15 weeks fetus was observed on the ninth postoperative day when the patient was discharged. Being a vertical scar and far-stay, the patient was willing to stay till suture removal. The patient is being monitored regularly and the fetus is healthy and growing well. The resected dermoid cyst of the ovary was diagnosed on the basis of the histopathological examination showing keratinizing, stratified squamous epithelial lining, intraluminal keratin, hair and sebaceous glands.The patient was kept on Intravenous tocolytics for firstDuring pregnancy, the most common ovarian tumors are serous cystadenomas, luteomas, corpus luteal cysts, and cystic teratomas . An appeOvarian torsion during pregnancy is rare and relatively little is known about the epidemiology of ovarian torsions, since only 1%-2% of pregnant women present with this condition, and most cases go undetected. Although most cases are asymptomatic, a large cyst can cause significant maternal and fetal morbidity rates if it ruptures or twists. Dermoid cysts are usually benign but may be involved with malignancy. To conclude, obstetricians should be alert to the possibility of acute ovarian torsion in expecting mothers and should be highly suspicious of it. Prompt surgical action benefits both the mother and the unborn baby.


Osteochondroma is a common benign bone tumor consisting of cartilage-covered bone confluent with the medullary canal of the epiphysis. Extraosseous osteochondroma shares the same appearance and histologic features as a typical osteochondroma but does not have any attachment to surrounding bone structures. Because of its low incidence, extraosseous osteochondroma is uncommon in clinical workups and thus prone to misdiagnosis. The diagnosis of extraosseous osteochondroma should be considered when there is a well-defined bony mass in the soft tissue with no direct continuity with the adjacent bone or joint. Here, we present a case of an imaging diagnosis of “calcified bursitis in the subcutaneous superficial fascial layer” and a postoperative pathological diagnosis of “extraosseous osteochondroma.”The patient was a 61-year-old man who had a right plantar heel mass for 2 years and recently visited the hospital because of discomfort in shoes.The patient was diagnosed with pathological examination.After completing the relevant preoperative examination and preoperative preparation and excluding contraindications to surgery, surgery was performed under nerve block anesthesia.We performed surgical resection, and the patient did not have obvious discomfort when discharged from the hospital. Auxiliary examination showed no abnormalities.For foot tumors, we need to consider the possibility of extraosseous osteochondroma. After completing the auxiliary examination, we should determine the relationship between the tumor and its surrounding tissues and blood supply before surgery to avoid causing major trauma. Osteochondroma is common and usually found in the epiphysis of long bones with a cartilage cap and continuation with the medullary cavity of long bones. Soft tissue osteochondroma is uncommon, which may be found in the synovial tissue of joints, tendon sheaths, or bursae. Only a few cases of extraosseous osteochondroma of the foot have been reported. Here, we report a case of extraosseous osteochondroma in the superficial fascial layer of the heel.The patient was a 61-year-old man who had a right plantar heel mass for 2 years and recently came to the hospital because of discomfort in shoes. On examination, the skin of the right heel plantar was not red, swollen, or broken. The skin was locally elevated, and a sub-circular swelling of around 2 cm in diameter could be palpated with unclear borders, tough texture, painful pressure, no obvious blood flow pulsation, and poor mobility. The foot and ankle were mobile with good peripheral sensation and blood flow. The patient underwent ultrasound examination at an external hospital, which showed that the plantar bursa was enlarged in the subcutaneous superficial fascial layer of the right plantar foot, and a strong echogenic mass was detected in the bursa with poorly defined borders, an irregular shape, and acoustic shadowing. The continuity of the Achilles tendon and plantar fascia of the right foot was intact, and no thickening was observed. Ultrasound suggested calcified bursitis in the subcutaneous superficial fascial layer of the right plantar foot. After admission to our hospital for surgical treatment, right heel X-ray was performed, which showed a patchy high-density shadow in the soft tissue of the lower edge of the right heel (Fig. After completing the relevant preoperative examination and preoperative preparation and excluding contraindications to surgery, surgery was performed under nerve block anesthesia, and a transverse incision of around 4 cm was made on the surface of the right plantar mass along the skin line Fig. , cut the,2 Therefore, clinical manifestations are not obvious; however, painful swelling and discomfort become more pronounced as the mass gradually increases in size.Extraosseous osteochondroma, also known as soft tissue osteochondroma, is a relatively rare, slow-growing benign soft tissue tumor that is usually found in the soft tissues of the hands and feet and presented as a small, discrete calcified mass rarely exceeding 2 to 3 cm in size. and the mass is not attached to the periosteum or bone cortex, nor is it within the confines of the joint capsule or tendon sheath. Computed tomography will likewise show the specific location and central calcification or ossification of extraosseous chondroma.,5 MRI is the best method to determine the boundaries of the mass, which shows a well-defined heterogeneous lesion with a mostly low signal intensity on T1WI and a mixed high and low signal intensity on T2WI. The T2 signal is moderately intense in areas of mature ossification and low in areas of high-density calcification.–6 In addition, MRI can be used to determine the size of the unsettled cartilage cap; the cartilage cap shows a low-to-moderate signal intensity on T1WI and a high signal intensity on T2WI.–6On imaging, the X-ray presentation of an extraosseous osteochondroma is a well-defined lobulated soft tissue mass with a central calcified or ossified area,,6–8The exact mechanism of the disease is unclear. Nevertheless, it is thought that the mesenchymal cells or fibroblasts in the interstitial matrix of the muscle tissue are stimulated by intrinsic or extrinsic factors, and they are transformed into chondroblasts or osteoblasts, gradually forming osteochondroma. Another hypothesis is the displacement of epiphyseal chondrocytes, resulting in new endochondral bone growth in the soft tissue. Some reports have suggested that it may be related to the chemotactic transformation of lipoma or endochondral osteogenesis.,9 Myositis ossificans is usually heterogeneous or amorphous in its early stages and can increase or decrease in size within a few weeks, typically presenting as a “banding phenomenon” of peripheral calcification consistent with muscle alignment and without the presence of a clear cartilage zone around the osteoclastic area.,10–12 Periosteal chondroma is found on the surface of long bones and can cause cortical erosion. It may also be confused with extraosseous osteochondroma on imaging. Tumoral calcific deposits typically appear on imaging as well-defined lobulated calcified masses that may be stratified on horizontal projection. The masses are easily distinguished by the presence of trabecular structures within them.,5,11,12In this case, the patient visited the hospital with discomforting heel plantar swelling and was diagnosed with calcific bursitis on imaging during the first ultrasound examination. Extraosseous osteochondroma needs to be differentiated from synovial osteochondromatosis, myositis ossificans, periosteal chondroma, and tumoral calcific deposits. Synovial chondromatosis is usually characterized by multiple cartilage and osteochondral nodules within the synovial membrane and can also manifest as intra-articular free bodies. Synovial chondromatosis is identified based on the presence of subsynovial cartilage growth.,5The diagnosis of extraosseous osteochondroma is difficult based on the patient’s clinical presentation and imaging examination, and a definitive diagnosis is largely dependent on the pathological histology. Clinicians should fully consider the relevant circumstances and focus on differentiation when encountering similar cases. After completing ancillary examinations, the relationship between the mass and the surrounding tissues and blood flow should be determined before surgical treatment to avoid causing considerable trauma. As there have been no reports of malignant transformation and metastasis, most patients are expected to have an ideal prognosis after surgical local excision. In a follow-up report by Chung and Enzinger, recurrence was identified as a potential problem, and surgical excision remains the first choice for 18% of recurrent patients.Shaobo Zhu collected the data, imaging, and operation reports and wrote the initial draft of the manuscript and subsequent revisions; Junhao Zeng was involved in editing and overseeing the text; Zhi Zhang was the chief surgeon of the patient; Cunmin Rong was the senior author and was responsible for oversight of the report and editing the manuscript.Investigation: Junhao Zeng.Resources: Zhi Zhang.Writing – original draft: Shaobo Zhu.Writing – review & editing: Cunmin Rong.


Huge primary epidural solitary fibrous tumors in the sacrum are a rare clinical entity. The purpose of this article is to present our experience in treating such large and complex neoplasms in a 31-year-old woman. The patient complained of constant nocturnal bilateral hip and lower back pain and unilateral radicular symptoms  in the left S1/S2 dermatomal distribution. Diagnostic imaging, biopsy, preoperative endovascular embolization, two-staged tumor resection, and lumbosacroiliac fusion were performed. The treatment resolved the patient’s neurological symptoms and resulted in overall good postoperative functionality. The patient has been in remission for more than five years despite her refusal of adjuvant radiotherapy. Solitary fibrous tumor (SFT), also previously known as hemangiopericytoma, is a rare mesenchymal neoplasm of fibroblastic origin, accounting for less than 2% of all soft tissue tumors . The 201A 31-year-old woman presented to the Department of Obstetrics and Gynecology in Vilnius University Hospital Santaros Klinikos for an elective laparoscopical diagnostic procedure due to a suspected ovarian cyst. She also complained of constant nocturnal bilateral hip and lower back pain and unilateral radicular symptoms  in the left S1/S2 dermatomal distribution that worsened over the last two years. No gynecological abnormalities were found, only a large retroperitoneal mass. Then she was referred to the Department of Neurosurgery in the same hospital where one of the authors gave further plan. Initial differentials involved more common masses in the sacral area, such as chordoma, chondrosarcoma, and giant cell tumor. There was no history of weight or appetite changes. Pelvic magnetic resonance imaging (MRI) was performed, which demonstrated a heterogeneously enhancing, multi-cystic, well-circumscribed tumor with pseudocapsule at the L5/S3 levels Figure .NAB2-STAT6 fusion was unavailable in our laboratory. The tumor fragments consisted of spindle-shaped cells with moderately atypical nuclei arranged in large cellular fascicles or palisades. Capillaries were neatly arranged, and no foci of necrosis or hemorrhage were observed. The mitotic index was <2 per 10 HPF. This information was of prognostic value when using the modified Demicco et al. system for predicting the risk of metastasis for the patient. Considering the age of the patient (31 years), the size of the tumor (12.0 x 10.0 x 9.3 cm), mitotic activity (<2/HPF), and the absence of necrosis in the histological view, this case should be regarded as a low-risk lesion with no risk of metastasis at 10 years. Immunohistochemical examination revealed a strong diffuse STAT6 and CD34 expression, moderately intense CD99, and weak alpha-smooth muscle actin staining; Ki67 proliferative index was around 4%. Epithelial membrane antigen (EMA) and S-100 protein were negative  and sexual life. The patient was discharged after surgical interventions, and further treatment was discussed with oncologists from the same hospital. She consciously declined the suggested option of adjuvant radiotherapy due to a desire to maintain her future childbearing potential. After a year and a half, she presented to the Department of Neurosurgery in Vilnius University Hospital Santaros Klinikos with renewed lumbago and pain in her left leg during movement. The spinal X-ray and pelvic CT confirmed broken right rod at the L5/S1 level. Revision was performed and both rods were replaced. Additional posterolateral bone fusion was performed using autologous spongious autografts from L4 and L5 spinous processes and bone substitute. The patient has been followed yearly for more than five years and no recurrence of the tumor was detected.Sacral SFTs are indolently growing and usually asymptomatic in early stages. However, they may cause compression symptoms ranging from radicular symptoms, lower back pain, to paresis, and less often bowel and bladder incontinence . It is iSurgery is a fundamental treatment, and even benign SFT tumors should be treated with wide surgical resection when possible . In someThis report described a unique case of huge primary epidural SFT in the sacrum. Before surgical removal, endovascular tumor embolization should be considered as these tumors are usually hypervascular. A two-staged piecemeal tumor resection through the combined anterior and posterior approach could be radical enough and safer than a single-staged surgery providing good control of the tumor and internal organs with surrounding structures. As a result, combined approach could preserve normal neurological function and good quality of life for the patient. To date, our patient has no signs of relapsed tumor despite her refusal of adjuvant radiotherapy.