Sacral giant cell tumors (GCTs) are rare entities that exhibit slow progressive growth and become clinically apparent when they reach a considerable size. The current case report discusses the presentation, investigation, and management of a young male patient diagnosed with a large sacral mass. A 17-year-old male patient presented with uremia, bilateral lumbar pain, and severe weakness of his lower extremities. Imaging revealed a midline sacral mass causing bilateral upper tract obstruction. The patient underwent bilateral nephrostomies followed by a partial en bloc sacrectomy and curettage of the tumor bulk. Histopathology revealed a giant cell tumor of the sacrum. Postoperatively, the patient received adjuvant radiotherapy and rehabilitation for his neurological symptoms. Sacral GCTs are essentially benign but behave like a malignant tumor in view of frequent recurrences and reports of malignant transformation. Surgery with wide local excision remains the ideal modality for complete clearance of sacral tumors. Nevertheless, limitations include their large size, difficult operative access, risk of fatal intraoperative bleeding, and inevitable high postoperative morbidity. Giant cell tumors (GCTs) of the sacrum are rare entities. This pathology mainly affects the long bones. In general, the incidence of giant cell tumors is reported at 1%-5% of all bone tumors and is mainly found in the long bones . The incSacral GCTs exhibit a silent progression of growth and become clinically apparent after they have reached a considerable size. Wide local resection in these cases imminently results in severely unacceptable neurological deficits. Despite management algorithms having been proposed, the ideal treatment for such tumors needs further deliberation.A 17-year-old boy presented to the neurosurgical department with new-onset urinary incontinence and long-standing bilateral lumbar pain along with weakness in his lower extremities. His past history revealed he had difficulty in walking, recurrent urinary tract infections, and worsening constipation for the past two years. On physical exam, he was wheelchair-bound, appeared pale and distressed. On abdominal examination, a non-pulsatile lower abdominal swelling was palpable, approximately 6*8 cm in size, which was non-tender to touch and had a firm to hard consistency. Furthermore, he had bilateral loin and sacral tenderness. On digital rectal exam, the rectum appeared to be displaced by a large swelling compressing the posterior wall of the rectum along with loss of anal tone and diminished perineal sensations. On neurological examination, there was a severe loss of power in both lower extremities (2/5) with bilateral absent ankle reflexes. His baseline investigations revealed abnormalities: hemoglobin of 9.0 mg/dl, serum creatinine levels of 5.4, and urea of 156 mmol/dl. Other lab parameters were within normal limits. Subsequently, an abdominal ultrasound revealed bilateral hydronephroureters and an ill-defined mass extending from the posterior surface of the bladder to the sacrum.A lumbosacral magnetic resonance imaging (MRI) scan (T1 -weighted) showed a large (10.7*10.0*8.3 cm) expansile lytic lesion, involving the midline sacrum with extensive soft tissue component and ill-defined margins, as shown in Figure Upon urological consultation bilateral nephrostomies were placed as a urinary diversion. Operatively, a highly vascular, friable, vacuolar mass arising from the sacrum was noted. Partial en bloc sacrectomy and subtotal resection of the tumor was performed due to per operative bleeding. The patient was transfused with four red cell units and two fresh frozen plasma units intraoperatively. The patient was referred to the regional oncology unit for radiotherapy and rehabilitation.The specimen histopathology report showed the following findings:Gross specimen: Multiple tan brown hemorrhagic tissue piecesMicroscopy: A tumor composed of sheets of the spindle to mononuclear cells with dispersed. multinucleated osteoclast-like giant cells. No atypia was seen. Rare mitotic activity observed.Immunochemical stains: CD68 (highlight giant cells), p53 (positive in giant cells).The long-term risk of recurrence and response to radiotherapy is yet to be established for our patient.Malignant and primary tumors of the sacrum are rare. Metastasis from distant sites, lymphoma, or multiple myeloma are more common as compared to primary sacral tumors. The differential diagnosis of sacral lesions identified on radiological imaging includes benign conditions such as giant cell tumor, aneurismal bone cyst, osteoid osteoma, osteoblastoma, and nerve sheath tumors. Malignant differentials may include sacral chordoma, chondrosarcoma, Ewing's sarcoma, osteosarcoma, and Paget's sarcoma.Classically, on CT and MR imaging, GCTs are known to be heterogeneous with areas of low attenuation that represent necrotic foci. Bleeding in the mass is demonstrated by a high signal intensity on T1-T2 weighting imaging. A low-intensity signal on T2 weighting represents areas of dense fibrosis and high hemorrhagic content within the neoplasm. Multiple fluid levels on imaging that represent hemorrhage and sedimentation characterize an aneurismal bony cyst. Osteoid osteomas are rare, with an incidence of 2% in the sacrum. They generally exhibit a low-intensity signal on T1 weighting and an intermediate-level signal on T2. A characteristic finding of osteoblastoma is usually a flare that is representative of an inflammatory response around the tumor, however, it is generally considered to have a nonspecific representation on MR. Hemangiomas of the sacrum depict a classical honeycomb appearance on imaging, as well as coarse trabecular thickening.Essentially, the management of sacral GCTs is surgical, non-surgical, or a combination of both. Surgical approaches include intra-lesional curettage, sclerotherapy, and subtotal or total en-bloc sacrectomy, whereas non-surgical options enlist selective arterial embolization (SAE), cryotherapy, radiotherapy, and, more recently, monoclonal antibodies.Selective arterial embolization of the sacral tumor-feeding arteries has shown promising results in terms of limiting the tumor progression of sacral GCTs. Hosalkar et al. published a series of nine patients of which seven showed no progression at a mean follow-up of 8.96 years [Radiotherapy and cryotherapy are other non-surgical modalities used for the treatment of sacral GCTs. Due to the high rate of local recurrence, these techniques are not preferable as a sole primary treatment. Leggon et al.  reportedThe treatment management of GCTs of the spine and sacrum is complex in nature. Surgical excision with wide local margins remains the ideal option for complete removal of the sacral tumor, with reports of a 0% risk of recurrence . NeverthThe management of sacral GCTs requires a multidisciplinary approach. Complete resection is the treatment of choice; however, radiotherapy serves as a suitable adjuvant therapy in cases of subtotal resection. Close monitoring is required to assess the risk of local recurrence and metastasis.


Diverticulosis of the small bowel is rare and, in most cases, discovered incidentally. However, diverticulitis and other complications are important to consider in the differential of an acute abdomen, especially in the elderly population.The patient was a 59-year-old female who presented with acute lower abdominal pain progressing to peritonitis. Computed tomography scan showed a large inflamed and perforated diverticulum on the mesenteric side of the jejunum. Exploratory laparotomy revealed a dilated proximal jejunum with a 5-cm inflamed and perforated mesenteric diverticulum. A small bowel resection with primary anastomosis was performed.Jejunal diverticulitis remains a diagnostic challenge. Although uncommon, owing to its high mortality rate, it is an important clinical entity to consider and requires timely management. Jejunal diverticulosis is a relatively rare condition with a reported annual incidence of 0.3–2.3% . The majA 59-year-old female presented to the emergency department with 48 h of sudden onset abdominal pain. The pain began across the lower abdomen and subsequently became diffuse involving all four quadrants. Associated symptoms included nausea without vomitus and fever. Her past medical history was significant for hypertension, type II diabetes mellitus, depression, and one episode of colonic diverticulitis managed with antibiotics alone. Vital signs were abnormal with a fever of 102 °F and tachycardia of 121 bpm, but normotensive 129/75 mmHg. On physical examination, the patient was tender to palpation throughout the lower and mid abdomen without rebound or guarding and had some fullness over the umbilical region.Laboratory results were notable for an elevated white blood cell count (15.3) with 81% neutrophilia. All other labs were within normal limits including blood urea nitrogen (BUN) (13), creatinine (0.92), and lactic acid (1.27).A CT scan of the abdomen with oral and intravenous (IV) contrast revealed a jejunal loop with a large diverticulum on the mesenteric side with associated diverticulitis and a 5.3 × 3.6 × 4.8 cm contained perforation. There was extensive edema of the entire jejunal loop and hazy infiltration of the adjacent mesentery Fig. a, b. No The patient was admitted with the diagnosis of jejunal diverticulitis with perforation but without clinical peritonitis. She was taken emergently to the operating room and underwent an exploratory laparotomy via midline incision. On entering the abdomen, a small amount of serous fluid was encountered without frank purulence. The involved jejunal loop was found to have serosal purulent exudates as well as interloop adhesions. The proximal jejunum was dilated and the distal jejunum collapsed. Twenty-seven centimeters of the proximal jejunum was resected to healthy non-inflamed edges, and a stapled side-to-side functional end-to-end anastomosis was performed. The small bowel was run from the ligament of Treitz to the ileocecal junction, and 4 other large non-inflamed diverticula ranging from 0.5 to 3 cm in diameter were seen along the mesenteric border. These were widely spaced and therefore not excised, as this would have required multiple further small bowel resections and anastomoses with associated increased morbidity. The abdomen was lavaged and closed.Gross examination of the specimen revealed a 27-cm length of the jejunum with focal areas of fibrinous material. Within the specimen were three diverticula originating from the mesenteric side of the jejunal serosa. On microscopic examination, one diverticulum demonstrated transmural inflammation with acute inflammatory exudate and perforation. The other two appeared non-inflammatory. The surrounding omentum was thickened with fibrinous exudates and focal areas of hemorrhage (Fig. Postoperatively, the patient’s course was uncomplicated. She had return of bowel function, tolerated a regular diet, and was discharged on postoperative day 6.Diverticular disease is a relatively common disorder and may be multifocal throughout the intestinal tract. Most frequently, it affects the colon; however, other sites include the jejunum, ileum, and duodenum. Jejuno-ileal diverticulosis was first described in 1794 by Somerling. It is relatively rare, comprising only 18% of all small bowel diverticula, which is likely an underestimate given they are often incidentally found on imaging –5. TheirTable In the majority of cases, jejunal diverticulosis is diagnosed incidentally either on imaging or intraoperatively. However, around 10–30% of patients present with disease complications including diverticulitis, perforation, bleeding, or small bowel obstruction . IsolateComplicated jejunal diverticulitis can be both a diagnostic and therapeutic challenge with a high mortality rate. We recommend that jejunal diverticulitis be considered a differential diagnosis in the acute abdomen and a CT scan will allow for the timeliest diagnosis. In cases of free perforation, operative resection of the affected segment with primary anastomosis appears to be a successful management strategy.


Perivascular spaces (PVSs) surrounding the walls of arteries, arterioles, and venules are a common finding in brain imaging. Even if they do not directly communicate with subarachnoid spaces, there are some cases in which subarachnoid hemorrhage (SAH) and intracerebral hematomas extend to the PVSs by leakage of the leptomeninges. In this report, we present a case of enlargement and bleeding of PVSs in the midbrain of a young woman with head trauma, without evident SAH or intracerebral hematomas. According to Hutchings and Weller  and OztüA 42-year-old woman, without previous disease, was admitted in the Emergency Room for a polytrauma during an accident car. Clinically she was conscious; she had a head trauma with hairy scalp; she complained headache and pain of the left foot; on the accident set, opiates were administered. Pressure of oxygen (PaO2) was 98%, cardiac frequency was 65 bpm, Glasgow Coma Scale was 15, and the neurological examination was negative. Laboratory tests revealed hemoglobin: 10.7 g/dL , white blood cell: 11.71 x 109/L , coagulation tests in normal range, and normal platelet count. A total body CT was performed; it demonstrated, beyond the hairy scalp with large soft tissue loss in frontoparietal zone, a multiloculated cyst in the left midbrain, with eccentric hyper-density, hematic-like , referring to scalp surgery. She complained headache, vertigo, nausea and diplopia, reflecting an involvement of the brainstem and the oculomotor nerves. A second CT and brain MRI with IV contrast were performed: CT scan showed a fluid-fluid level, with enlargement of left cerebral peduncle and a slight mass effect Figure . No neurTwo days after the surgery, diplopia and nausea were disappeared, vertigo persisted. The patient stayed six days more in hospital to follow the reconstruction of the scalp and to recover. Unfortunately, the patient had a reject of transplanted skin with necrosis and had to schedule a new surgery in a month later. Before discharge, a CT scan was performed in order to assess the midbrain lesion Figure .The patient was discharged and she continued the treatment for the scalp lesion and gradually recovered from vertigo. She repeated CT and MRI exams to follow the midbrain lesion up: a CT scan after one month Figure  and a MRThe PVSs were described for the first time by Rudolf Vircow and Charles Philippe Robin, in 1851 and 1859 respectively -7. So faA recent review discussed the role of mild traumatic brain injury as a cause of VRSs pathological enlargement . Even inIn the end, we can speculate about possible differential diagnosis of our case. In 2007 a review described PVSs at MRI , exhibitThis case shows enlargement of PVSs in the midbrain of a young woman with head trauma, with a peculiar bleeding only inside these PVSs. In literature, there are some cases reporting bleeding of PVSs, but they were associated with the presence of SAH and intracerebral hematomas, extending to the PVSs by leakage of the leptomeninges. To our knowledge, this is the first report of enlargement and bleeding of PVSs in the midbrain after brain injury, without evident SAH or intracerebral hematomas.


The concomitant occurrence of duodenal atresia (DA) and a choledochal cyst (CC) has rarely been reported. Knowledge of both the presentation and management of this rare co-occurrence is imperative in avoiding potential complications and sequelae, such as biliary metaplasia. Herein we describe a female infant born at 32 weeks gestational age who was diagnosed with duodenal atresia and annular pancreas postnatally, who had subsequent findings of malrotation and a choledochal cyst, as seen from contrast imaging. Uncomplicated repair of the DA and obstruction was performed at 4 days of life. She re-presented 2 years later with non-bloody, nonbilious emesis and was found to have elevated amylase, lipase and liver enzymes. Imaging revealed dilated intra-hepatic ducts, a distended gallbladder and a large choledochal cyst. She underwent a cholecystostomy tube placement followed by a definitive choledochal cyst excision with immediate improvement following surgery and full resolution of symptoms before discharge. Congenital obstruction of the duodenum can be complete or partial and due to an intrinsic or extrinsic etiology. Intrinsic obstruction is often caused by duodenal atresia (DA) and occurs in approximately 1 in 5000 to 1 in 10,000 live births . The conA female infant weighing 1700 g was born at 32 weeks gestational age to a 42-year-old mother. The pregnancy was complicated by gestational diabetes, with a non-immune rubella titer and prenatal ultrasound (US) revealing polyhydramnios and an echogenic bowel. Imaging after birth revealed a double-bubble sign typical for duodenal atresia.  A subseqAt two years of age the patient presented to the emergency department with feeding intolerance, persistent emesis, elevated liver transaminases , elevated amylase >1446 U/L and lipase 465 IU/L. Magnetic resonance imaging (MRI) revealed significantly dilated intra- and extra-hepatic ducts, a distended gallbladder and a large choledochal cyst. The physical exam was significant for ascites and jaundice. She underwent an urgent laparotomy, which found bilious ascites requiring subsequent placement of a cholecystostomy tube. A cholangiogram was then performed due to the persistent feeding intolerance. This re-demonstrated the intra- and extra-hepatic ductal dilation and incomplete biliary obstruction.  She undeThis case demonstrates a case of antenatally formed DA and CC with concomitant anomalies requiring surgical intervention due to an obstructed hepatobiliary system 2 years after the initial findings. The embryologic origin of the duodenal atresia centers on the failure of the recanalization of the duodenum after the 7th week of gestation and can be associated with other congenital anomalies such as Down’s syndrome, which is present in 25–40% of cases . Other aSimilarly, pediatric cases reported in the literature since the early 1990s have included findings of PBM associated with duodenal obstruction and subsequent choledochal cysts. The first case of CC and its surgical removal was reported by Bailey et al. in 1993 . Iwai etThis same finding was described by Iwai et al. involving a 37-week gestational age female infant born with DA repaired at birth, who presented at 4 years of age with jaundice, vomiting and was found to have a choledochal cyst and dilatation of the left intrahepatic duct requiring surgical resection . Iwai thDespite the rarity of this association, the proposed mechanisms have all seemingly involved an anomalous formation of the pancreaticobiliary junction. Early case reports have nearly identical imaging findings, including an anomalous pancreaticobiliary ductal junction found during the intraoperative cholangiogram, annular pancreas and CC ,9,10. ThThe simultaneous occurrence of congenital duodenal atresia and a choledochal cyst, although rare, have common anatomical findings in all case reports throughout the years. These findings offer an underlying connection between an anomalous arrangement of the pancreaticobiliary system, duodenal obstruction, annular pancreas and the gradual dilation of bile ducts into symptomatic choledochal cysts. This emphasizes the importance of early evaluation in infants presenting with DA, to identify abnormalities such as CC and annular pancreas to prevent the development of dangerous sequelae, such as chronic cholangitis, pancreatitis, biliary metaplasia or the need for invasive surgical interventions in later childhood.No financial relationships relevant to this article to disclose.


Abdominal ventral hernias are common in chronic liver disease due to increased abdominal pressure and sarcopenia. Following liver transplantation, diagnosis of chronic abdominal pain is challenging because it may relate to immunosuppression, scaring or opportunistic infections.A 62 years-old male presented with chronic abdominal pain one year following liver transplantation due to hepatocellular carcinoma. After work-up he was diagnosed with a Spigelian hernia containing the appendix. We did hernia repair with mesh but appendectomy was not performed since it showed no signs of inflammation. On follow-up the patient had complete resolution of the pain.This is the first case of spigelian hernia containing the appendix following liver transplantation. Mesh repair can be safely performed in this setting but incidental appendectomy is controversial due to higher morbidity and mortality. In this case report we discuss the relationship between liver transplantation, abdominal hernias and the pitfalls of incidental appendectomy.Uncommon ventral hernias are a possible cause for chronic abdominal pain after surgery and should be investigated with imaging studies. Mesh repair is safe but incidental appendectomy in the immunosuppressed is not encouraged due to increased morbidity. Sarcopenia and increased abdominal pressure in cirrhosis play a role in their development and may persist following transplantation . Up to 4Early diagnosis is challenging since symptoms can be subtle and chronic abdominal pain may relate to adhesions, immunosuppression or opportunistic infections. Transplantation may induce metabolic syndrome and weight gain, making physical examination more challenging. Also, due to large incision and immunosuppression, patients are less responsive to painful stimulus. Therefore, diagnosis of abdominal pain post liver transplant is not straightforward and it is important to be aware of differential diagnoses and uncommon clinical scenarios.This case report has been reported in line the SCARE criteria  and it a2A 62 years-old male with long-term diabetes, hypertension, dyslipidemia and non-alcoholic fatty liver disease (NASH) was diagnosed with hepatocellular carcinoma. His past surgical history included a previous right inguinal hernioplasty with mesh. After complete work-up he was deemed eligible to liver transplantation and surgery was uneventful.On follow-up the patient was placed on immunosuppression with tacrolimus and remained the use of aspirin, metformin, atorvastatin and gliclazide for control of his clinical comorbidities. During office visits in the first year following surgery, he complained of chronic right-sided abdominal pain exacerbated by movement. Clinical examination was unremarkable and no hernias were detected on previous incisions. We performed a complete medical work-up and abdominal computed tomography that revealed a spigelian hernia containing the appendix . This fiThe patient was listed for elective surgery, which was performed by the authors LFS and LE. At the operation, an oblique incision was made and after opening the skin and subcutaneous, a bulge just below the fascia of the external oblique was noticed . Opening3To our knowledge this is the first case of spigelian hernia (SH) containing the appendix after liver transplantation, which could result in higher morbidity and mortality if not promptly diagnosed. Previous reports included mostly patients with acute complications, such as incarceration and acute appendicitis ,7. In onSH was described in 1764 by Josef Klingosh and named after the anatomist Adrian Spieghel. The true prevalence is uncertain but may be up to 2% of abdominal hernias. The apparent incidence has increased over the last decades, possibly due to the increased incidence of obesity and the accuracy of diagnostic imaging . SH diagThe content of the hernia sac is variable: omentum, small bowel, colon, epiploic appendage have all been described. SH containing the appendix is a rare entity and only few cases have been reported in the literature. This hernia type lacks an eponym and is considered a different entity from Amyand hernia, which corresponds to an inguinal defect with the appendix.Incisional hernias after liver transplantation are not uncommon and have been reported in up to 43% of patients . FikatasIncidental appendectomy remains controversial but it is reasonable to operate on young and fit patients with low morbidity. However, it is not recommended to operate on the elderly and the immunosuppressed due to increased risk of complications and the lower chances of developing acute appendicitis in older age. Kotaluoto et al. analyzed appendectomies performed for over two decades in Finland and observed a 39-fold increase in mortality among the elderly .It is important to notice that acute appendicitis in immunosuppressed patients will often not present with the most common symptoms and the clinical picture may be misleading. Fortunately, our patient was diagnosed before any complication ensued.4Chronic abdominal pain following liver transplantation is a challenging diagnosis. Uncommon ventral hernias are a possible cause for chronic abdominal pain after surgery and should be investigated with imaging studies since physical examination alone is unreliable. In the context of immunosuppression, mesh repair is safe but incidental appendectomy is not encouraged due to increased morbidity and mortality.There is no relevant conflict of interest.We did not receive any funding for this case report.Ethical approval exemption was given for this study.Written informed consent was obtained from the patient.Lucas Faraco Sobrado: writing - original draft, review and editing.Lucas Ernani: writing - original draft, review and editing.Daniel Reis Waisberg: writing - original draft, review and editing.Luiz Augusto Carneiro D’Albuquerque: writing - review and editing, supervision.Wellington Andraus: writing - review and editing, supervision.Not applicable.Lucas Faraco Sobrado.Not commissioned, externally peer-reviewed.


Mesenchymal stem cells differentiate into a plethora of cell types. These differentiated cell types include osteoblasts, chondrocytes, myocytes, and adipocytes. Sarcomas occur secondary to malignant transformation of these mesenchymal, pluripotent stem cells. Involuntary smooth muscle is responsible for the make up of hollow organs and vasculature in our body and is regulated by our autonomic nervous system, hormones, as well as chemical and local mediators. Cancer involving smooth muscle cells is designated as leiomyosarcoma (LMS). LMS can arise from any location in the body where smooth muscle is present and is frequently reported in the abdomen and pelvis, as well as the trunk, extremities, and throughout the retroperitoneum.  Sarcomas represent less than a fraction of a percent of overall malignancies . They arLeiomyosarcoma (LMS) occurs secondary to malignant transformation of smooth muscle cells in our body. The exact etiology of LMS is unknown, however, like many other cancers, genetics and environmental factors can likely be attributed. LMS is differentiated from its counterpart, leiomyoma, with radiologic and histopathologic evaluation (HPE). The presence of cellular atypia, high mitotic rate and areas of focal necrosis on HPE favor a diagnosis of LMS. On magnetic resonance imaging (MRI), LMS will often display nodular borders, inhomogenous enhancement with intralesional vessels and hemorrhage on T2-weighted images, with hypervascularization, peripheral early enhancement, and central necrosis [According to an epidemiological study by the United States National Cancer Database from 2002-2014, the Surveillance, Epidemiology and End Results (SEER), found that of the 78,527 patients with a diagnosed sarcoma, 11,487 were LMS, making it the most commonly reported soft-tissue sarcoma diagnosed, followed by malignant fibrous histiocytoma 5,703), and liposarcoma . The top three reported primary tumor sites in this study included soft tissue , abdominal viscera , and thoracic viscera  [, and lipRetroperitoneal sarcomas are commonly diagnosed late in the disease course as patients are often asymptomatic until the tumor begins obstructing nearby structures. According to studies at Memorial Sloan Kettering, New York, the five-year survival rate of patients with locally-advanced LMS at diagnosis is largely dependent on the ability to be surgically resected. With complete resection and clear margins, the five-survival rate approaches 60%. With partial resection or unclear margins, the five-year survival is roughly 35% [We present a case of a 50-year-old female with a previously diagnosed LMS of the left renal vein that extended throughout the IVC eventually terminating in the right atrium who continued to have disease progression despite undergoing chemotherapy treatment.Our patient is a 50-year-old female that presented to our emergency department after suffering from a gradual onset of symptoms including dyspnea on exertion, left-upper quadrant abdominal pain, left-sided flank pain, and decreased appetite for a duration of two weeks.Prior to arriving at our institution, our patient had a biopsy proven diagnosis of a left renal vein LMS made six months earlier and had since been undergoing chemotherapy treatment at a neighboring hospital. The patient finished an eight-week course of doxorubicin, gemcitabine, and docetaxel anhydrous two-weeks before presenting to us. An interval computed tomography (CT) scan of the chest, abdomen and pelvis was performed following this chemotherapy course which demonstrated a large thrombus in the IVC not appreciated on prior imaging. Staging at this point was documented as T3N0M0. The patient had been informed that there were no further chemotherapeutic options left available to her and so she began investigating surgical options which is when she established care with our own surgical oncologist. The patient notified our surgical oncologist of her new onset symptoms and it was recommended that she go the emergency department and be admitted for further evaluation due to the nature of her complaints in this context.Vitals signs in the emergency department remained stable. The patient was afebrile with a temperature of 98.6 degrees Fahrenheit, a respiratory rate of 18 breaths per minute, a left-arm seated blood pressure of 120/65 mmHg, a pulse rate of 88 beats per minute, and bedside pulse oximetry of 98%. Complete cell counts (CBC), complete metabolic panels (CMP), coagulation profiles, and a urinalysis were obtained. Initial CBC was significant for a moderate anemia with a hemoglobin of 8.7 g/dL (12.0-16.0 g/dL) for which she was being treated with epoetin alpha. The CMP, coagulation profile and urinalysis were unremarkable. During the hospital course, the patient underwent numerous imaging studies. Initial chest radiograph Figure  showed nNo segmental mismatched perfusion defects were identified in either lung on ventilation-perfusion scan, representing a very low probability for a pulmonary embolus. Duplex ultrasound of the lower extremities resulted negative for deep vein thrombosis bilaterally. Right-upper quadrant (RUQ) doppler ultrasound was ordered to assess the liver, gallbladder, and IVC. An echogenic thrombus within the proximal IVC was seen Figure , 3.In addition, the main portal vein displayed pulsatile flow Figure , often sA nuclear medicine triple-phase renal scintigraphy was also performed to assess the degree of renal involvement and functional status. The results showed evidence of increased flow and secretion in the right kidney when compared to the left. The left kidney demonstrated decreased excretion within the excretory range which can be indicative of mild obstruction versus tubular injury.A CT scan of the chest with contrast showed no evidence of metastatic disease. Repeat CT scan of the abdomen and pelvis with contrast demonstrated a large inhomogenously enhancing left retroperitoneal mass arising from the left renal vein with invasion of the left kidney and accompanying intruluminal extension into the left gonadal vein, and IVC Figure , 6. To further evaluate the mass evident on CT abdomen with contrast, an MRI scan of the abdomen and pelvis with and without contrast was ordered. On the MRI, the mass measures 10.2 x 12.7 x 14.8 cm . The mass was characterized as T1 hypointense, T2 heterogenous and mostly hyperintense. The apparent epicenter is visualized at the left renal vein where the most avid area of enhancement is noted exerting a mass effect on the left kidney and invades the renal sinus venous structures  in greater than 70% of cases, and less frequently stain positive for cytokeratin and epithelial membrane antigen (EMA) in roughly 40% of cases .Imaging findings of retroperitoneal LMS on CT and MRI scans are nonspecific. Similarly to other sarcomas on MRI, retroperitoneal LMS appear to have decreased signal on T1-weighted imaging and increased signal on T2-weighted imaging . AlthougThe prognosis and five-year survival rate of LMS strongly correlate with factors such as if the tumor can be surgically resected, tumor location, and if local extension and/or metastases are present. Surgical resection is known to be the standard of care for therapy in LMS. However, there is no consensus on standard radiation and chemotherapeutic regimens for retroperitoneal LMS. A case reported by Sharma et al. reported success using doxorubucin, ifosfamide, dacarbazine, and mesna combined with radiotherapy, however the renal LMS measured only 3.8 x 3.6 cm and was designated as low-grade , in contNo randomized control trials have demonstrated the efficacy of radiation and chemotherapy in locally-advanced or metastatic renal LMS cases, and therefore this topic warrants further investigation for patients who are not surgical candidates or continue to have disease progression while undergoing chemotherapy, as seen in our patient. However, for localized renal LMS cases, current experimental evidence suggests against the use of radiotherapy as a neodjuvant option. The STRASS I trial, an open-label, randomized, phase 3 study conducted on 266 patients with histologically documented, localized, primary retroperitoneal sarcoma demonstrated no survival benefit with neoadjuvant radiotherapy prior to surgery . The STRThe risks and benefits of adjuvant, neoadjuvant, and surgical therapy should be considered carefully in the context of each patient, as previous case reports have mentioned favorable outcomes, others have not. Retroperitoneal LMS typically require surgical resection, chemotherapy, and in certain cases targeted radiation. Histologic and radiologic evaluation are vital aspects of the diagnostic workup and aid in creating individualized treatment plans. As previously mentioned, this malignancy is often diagnosed late in the disease course, when a patient experiences symptoms secondary to mass effect such as abdominal pain, cramping, and early satiety and/or loss of appetite. Our case demonstrates the need for multi-specialist care teams in order to give patients who are unfortunately diagnosed with a retroperitoneal LMS the best outcomes.


Glomus tumors (GTs) are mesenchymal neoplastic lesions arising from the glomus bodies and generally occur in the fingers and toes. Gastrointestinal GTs are rare, and most of them originate from the stomach; however, GT arising from the duodenum is exceedingly rare.A 68-year-old man was admitted due to abdominal pain. Endoscopy showed a round, smooth, elevated mass in the second portion of the duodenum with central ulceration. Abdominal contrast computed tomography showed a hypervascular tumor measuring 26 mm in diameter in the second portion of the duodenum, and pancreatic invasion was suspected. Endoscopic ultrasonography of the lesion confirmed a hypoechoic mass arising from the fourth layer of the duodenal wall. A biopsy was performed for central ulceration, and immunochemical studies showed positive results for smooth muscle actin (SMA) and negative results for S100, C-Kit, and CD34. Leiomyoma or gastrointestinal stromal tumor was suspected and pancreatoduodenectomy was performed. The specimen exhibited a vascular-rich tumor, 24 × 24 × 19 mm in size, with deep ulceration in the duodenum. Histological examination showed uniform small round cells with central nuclei and a pale cytoplasm (glomus cell) with perivascular proliferation. Immunochemical studies showed that the tumor was positive for SMA and collagen type IV, and negative for C-Kit, CD34, desmin, and S100. We diagnosed the tumor as a GT of the duodenum.GTs of the duodenum are exceedingly rare, but should be considered in the differential diagnoses of duodenal submucosal lesions. Glomus tumors (GTs) arise from the glomus bodies that act as skin thermoregulators, and are mostly found in the peripheral soft tissues at the dermal and subdermal subungual zones of fingers and toes . Among tA 68-year-old man presented to his primary care physician with a complaint of abdominal pain. He had a medical history of reflux esophagitis and benign prostatic hyperplasia. Esophagogastroduodenoscopy was performed, which showed a submucosal mass in the second portion of the duodenum with central ulceration. Upon admission, his carcinoembryonic antigen (CEA) and carbohydrate antigen (CA) 19-9 levels were within normal limits.Dynamic CT showed a circumscribed tumor measuring 26 mm in diameter in the second portion of the duodenum with ulceration. On the arterial and portal phase contrast-enhanced CT, the mass was greatly enhanced Fig. a, b withThe T1-weighted image showed the tumor as a hypo-intensity Fig. a, and thEndoscopy showed a round, smooth, elevated mass in the second portion of the duodenum with central ulceration Fig. a. EndoscPreoperatively, we diagnosed the tumor as a leiomyoma or gastrointestinal stromal tumor (GIST). Therefore, after informed consent was obtained, pancreatoduodenectomy (PD) was performed.The resected specimen showed a yellowish-white tumor, 24 × 24 × 19 mm in diameter, arising from the submucosa of the duodenal wall with deep ulceration  in alveolar hyperplasia around the small vessels. Immunohistochemical findings are useful for distinguishing GT from other subepithelial tumors. GT tumors are positive for SMA, vimentin, and collagen IV. In contrast, negative for C-Kit, CD34 (rarely positive), desmin, S100, and CD45 . In our Treatment of duodenal GTs consists of endoscopic mucosal resection, laparoscopic wedge resection and PD Table . PreoperWe present a rare case of GT in the duodenum. Duodenal GTs are difficult to distinguish from other submucosal lesions in the duodenum; however, immunohistochemical staining helps determine the correct diagnosis, and dynamic CT aids in preoperative diagnosis. Furthermore, GTs of the duodenum are exceedingly rare, but should be considered in the potential diagnosis of a duodenal submucosal lesion.


Supplemental Digital Content is available in the text Spinal involvement in adult Langerhans cell histiocytosis (LCH) is rare, and epidural involvement is unusual. LCH is mostly indistinguishable from other spinal lesions such as infection, lymphoma, and metastasis. So, it could be easily misdiagnosed without suspicion.We report a case of a 33-year-old man who complained of gait disturbance with weakness in both legs and severe back pain.A continuous enhancing epidural lesion with cord compression from the T7 to L1 level was detected in magnetic resonance imaging. Laboratory analysis indicated the possibility of spinal infectious disease. We assumed that the lesion could be tuberculous spondylitis.The patient underwent posterior laminectomy with marginal excision of the epidural mass to relieve cord compression. Pathological examination confirmed the diagnosis of LCH. The 12-month follow-up evaluation revealed that the patient was neurologically intact and had no gait disturbance.This case report presents a patient with epidural LCH of the thoracic spinal cord, which can mimic spinal infections such as tuberculous spondylitis with abscess formation. Therefore, LCH could be considered as a possible diagnosis when a patient presents with features of infectious spondylitis with vertebral involvement. The disease varies widely in clinical presentation from the localized involvement of a single bone to a widely disseminated life-threatening disease. Spinal involvement in adult LCH is rare, and epidural involvement is unusual.–5 LCH is mostly indistinguishable from other spinal lesions such as infection, lymphoma, and metastasis. Here, we report a case of a 33-year-old man diagnosed with epidural LCH of the thoracic spinal cord, which was confusing as clinical, laboratory, imaging findings indicated a spinal inflammatory disease.Langerhans cell histiocytosis (LCH) is associated with the clonal proliferation of Langerhans cells occurring as an isolated lesion or as part of a systemic  proliferation.2Informed written consent was obtained from the patient for publication of this case report and accompanying imagesA 33-year-old man was presented with 1 year of lower thoracic back pain, 7 days of gait disturbance, and weakness in both lower extremities. He also complained that he had difficulty maintaining his balance. There was no history of systemic illness and family history. On examination, the deep tendon reflex of both knees was exaggerated, and the motor grade of both lower extremities  was decreased. Blood test variables including white blood cells (WBC), erythrocyte sedimentation rate (ESR), and C-reactive protein (CRP) were increased . His body temperature was normal, and there was no inflammatory focus in other systems on clinical examination.Magnetic resonance imaging (MRI) showed a continuous enhancing epidural lesion, and computed tomography (CT) showed multiple osteolytic bone lesions at the T7–L1 level with spinal cord compression at the T9–T12 level  is more common than single system involvement (31%) at the time of diagnosis. In single system involvement, LCH mainly involves the lungs (51.1%), bone (38.3%), and skin (7.01%). In cases involving the bone, the location of lesions includes the skull (27%), femur (15%), and spine (6.5%). In our case, the patient was diagnosed with LCH as an adult, and the lesions involved the spine but not the lungs, skull, and appendicular skeleton.LCH is a rare disease with a rather unpredictable course as it can spontaneously resolve or progress to a disseminated form, compromising vital functions with occasionally fatal consequences. LCH is diagnosed mostly in children and adolescents and rarely in adults. The peak incidence is between the ages of 5 and 10 years in about 80% of cases. The lesions are usually solitary, and the vertebral bodies are the main structures affected.,4,6,12 The most common radiographic features in spinal LCH are lytic lesions of the vertebra, which can usually lead to vertebral collapse. In our case, there were altered signal intensities with enhancement in the vertebral bodies; however, the vertebral height was relatively intact. MRI revealed a continuous enhancing epidural lesion with spinal cord compression. Huang et al have reported that the typical signs of LCH are seldom observed in adults (11%), and epidural cord compression with neurologic symptoms is more common in adults than in children. The relatively preserved disc space as well as the multiplicity of vertebral body involvement and paraspinal and epidural enhanced lesions may suggest the possibility of tuberculous spondylitis.–16Vertebral lesions occur most commonly in the thoracic spine and less often in the lumbar and cervical spine.,6,12,17In our case, WBC, ESR, and CRP were sufficiently elevated to suggest the possibility of spinal infectious disease. Hence, we wrongly assumed that the lesion could be tuberculous spondylitis based on MRI findings and laboratory results. It is well known that LCH involvement can lead to the elevation of inflammatory markers such as WBC, ESR, and CRP. However, in previous studies related to adult LCH, results on inflammatory markers were absent or reported as normal.,19 To the best of our knowledge, there are no such reports for adults.The patient underwent posterior laminectomy with marginal excision of the epidural mass to relieve cord compression. The preoperative impression was tuberculous spondylitis with abscess formation; however, intraoperative findings did not correspond to an infection. The epidural mass had a whitish color and hard rubber-like consistency. Pathological examination confirmed the diagnosis of LCH. Similar to our case, the misdiagnosis between tuberculosis and LCH has been reported in some cases involving children.In MRI, epidural LCH is usually indistinguishable from other spinal lesions such as infection, lymphoma, and metastasis. Laboratory and clinical findings often lead to an inaccurate differential diagnosis. Ultimately, a definite diagnosis can be established by pathological confirmation. Therefore, diagnosis and treatment should not be concluded before evaluating biopsy results. Given the different possible causes, a diagnostic modality for tissue confirmation such as CT-guided biopsy should be performed. Chemotherapy may be used as a first-line therapy for patients without neurological deficits or as an adjuvant therapy after surgery, especially in cases with multiple bone lesions such as in the present case. Patients with evident signs of spinal cord compression, such as in our case, should promptly undergo surgical decompression to improve the chances of neurological recovery and to obtain tissues for histopathological examination. Regarding an optimal first-line chemotherapy agent, some experienced clinicians prefer to start with vinblastine and prednisone. However, vinblastine and prednisone have not been proven effective for adults in a prospective study. Due to a lower risk of neurotoxicity and unacceptable steroid-induced side effects, some experts prefer monotherapy with cladribine, cytarabine, or etoposide. In our case, the patient underwent surgical decompression and postoperative chemotherapy with vinblastine and prednisone.The optimal treatment strategy for spinal LCH remains controversial, and several treatment approaches including bed rest, immobilization with cast and brace, chemotherapy, radiation therapy, and surgery have been proposed. Nevertheless, the appropriate treatment options for adults have not been clarified by clinical trials and the literature. Some recommendations have been proposed for the evaluation and treatment of adult patients with LCH.In summary, we report a case of adult epidural LCH with spinal cord compression mimicking spinal abscess due to Mycobacterium tuberculosis. Although extremely rare, LCH should be considered when there are epidural lesions with spinal cord compression in adults.Conceptualization: Jae Hwan Cho.Data curation: Cheong-Su Lim.Formal analysis: Cheong-Su Lim.Investigation: Cheong-Su Lim.Methodology: Jae Hwan Cho.Supervision: Jae Hwan Cho.Validation: Jae Hwan Cho.Writing – original draft: Cheong-Su Lim.Writing – review & editing: Jae Hwan Cho.


Lipomas are frequently presented in adults and account for almost 50% of all soft-tissue tumors. Osteolipomas are rare and usually located in the intraosseous region or adjacent to bone. It is very unusual for osteolipomas with no connection to bony structures. We report a rare intramuscular osteolipoma independent of bone tissue.We report a case of a 58-year-old man with painful and progressively enlarging mass in the right lower leg. A plain X-ray and computed tomography (CT) scans revealed a large homogeneous, low-fat density mass containing an oval shape calcification without bone connection. MRI showed a circumscribed mass in the peroneus muscle with a large calcified component. The patient underwent surgical excision of the mass. Histologically, benign osteolipoma was the final diagnosis. No recurrence was observed at six months follow-up.Lipoma is a common benign soft tissue neoplasm but osteolipoma is rare. Most cases osteolipomas are connected with bone. independent of bone tissue has been reported in very few cases. Most of them occurred in the head and neck area. The pathogenesis of osteolipoma is still not clear. Although CT and MRI are useful for differential diagnosis, care should be taken because sometimes they are indistinguishable from well-differentiated liposarcomas. Excisional biopsy is useful for definitive diagnosis.Although ossifying lipomas are very rare, it is important to keep them in mind when a lesion with adipose tissue in combination with ossification is encountered. Varia2A 58-year-old man presented to our hospital with a painful and progressively enlarging mass in the right lower leg. The patient noted a soft tissue mass in the anterolateral aspect of his lower leg seven years before. He had no family history, no medical history and had not any severe trauma or irradiation to the region. One month prior to presentation, his discomfort became obvious and he began experiencing pain and tenderness. The pain was dull-aching in nature and did not radiate to other regions.On physical examination, a giant mass that was ovoid, firm, tender, well demarcated, and relatively fixed was palpated in the right peroneus muscle upper area and size was about 10 cm length and 2 cm width. The knee movements was normal. The neurological examination was within normal limits, and no lymphadenopathy was present. Laboratory data showed normal values including calcium, phosphorus, and alkaline phosphatase.A plain X-ray and computed tomography (CT) scans revealed a large homogeneous, low-fat density mass containing an oval shape calcification from fibula neck to fibula shaft about 11 cm length and 2 cm width. Continuity between a tumor and fibula was not found. . MagnetiAn excisional biopsy was undertaken. During the operation, a well encapsulated tumor mass was found to be located in the peroneus muscle . There wGrossly, the resected specimen is a well demarcated, diffusely yellowish adipose tissue mass measuring 11 × 3.5 × 2 cm and 43 g in weight. Outer surface is diffusely smooth because of a thin fibrous capsule. On sectioning, the cut surface consists of deep yellowish fat admixed with multiple scattered calcified white or brown bone .Fig. 4GrThe microscopic analyses revealed osseous trabeculae inside a mature adipose tissue. No cellular atypia or increased of mitotic figures were observed. These findings led us histopathological diagnosis of benign osteolipoma .Fig. 5HiThe definitive pathological diagnosis was intramuscular osteolipoma without evidence of malignancy. No recurrence was observed at six months follow-up.3Lipoma is a common benign soft tissue neoplasm that sometimes may have mixed tissue components. Lipomas with mixed components are named according to the type of tissue. Ossification of a lipoma was first described in 1959, and it is rarely reported . In a seThe pathogenesis of osteolipoma is still not clear. Two main theories exist for the pathogenesis of osteolipomas ,5. FirstCongenital malformations of bones, benign tumors containing bony tissue , secondary ossification due to trauma, liposarcoma with metaplastic changes should be considered in the differential diagnosis. The use of CT scanning provides excellent visualization of the calcified or ossified components of a lipoma and confirmation of proximity to adjacent bone, and MR imaging can also provide detailed information that is useful for further evaluation ,17. Howe4Although ossifying lipomas are very rare, it is important to keep them in mind when a lesion with adipose tissue in combination with ossification is encountered.No funding was requested.This is a case report study, no ethical approval was needed. On the other hand, the patient had been informed and gave their consent regarding this publication.Written informed consent was obtained from the patient for publication of this case report.JH Han performed the operation and perioperative management of the patient. JH Han also acquired and interpreted the data and drafted the manuscript. SM Hwang participated in the operation, perioperative management of the patient. S Choi revision of the manuscript. KR Sohn reviewed pathological findings.This manuscript is not a human study, but a case report.Jae Hwi Han.Not commissioned, externally peer-reviewed.No conflict of interest for all authors.


Myxofibrosarcoma (MFS) is a rare subtype of a malignant soft tissue tumor that occurs mainly in adults, and peaks at the age of 70. It typically presents as a slow growing, painless mass in the proximal part of the extremities. It is characterized with a high recurrence rate and a low rate of distant metastases; the most common metastases site is the lungs, and in some extremely rare cases it was mentioned that there was metastases to the head and neck region. We here report the first case of a myxofibrosarcoma metastasized from the gluteal region to the pterygopalatine fossa, which is the first report in the literature of this rare metastatic spread of myxofibrosarcoma.a 70 year-old male presented with diplopia and limited right eye movement. His medical history was significant for myxofibrosarcoma in his gluteus maximus. Magnetic resonance imaging showed a low signal mass in the pterygopalatine fossa. The tumor neither invaded the maxillary bone nor the maxillary sinus; the therapy plan was resection of the mass by partial maxillectomy followed by adjuvant radiotherapy.Metastasis to the pterygopalatine fossa should be considered in a patient with myxofibrosarcoma history presents with neuro-opthalmic symptoms. Partial maxillectomy in tumors that do not infiltrate into adjacent structures should be considered as a minimally invasive therapy. •Myxofibrosarcoma can rarely metastasize to the pterygopalatine fossa from the lower limb.•Developing diplopia after lower limb sarcoma may refer to metastasis to the head.•Less invasive interventions are recommended when no bone involvement is present.•Good patient consult and psychological support is essential for optimal outcomes. It was first described in 1977 by Angervall et al. , and wasIn this report, we are presenting a rare metastatic spread of MFS from the left gluteus to the pterygopalatine fossa. To the best of our knowledge, this is the first article that reports this rare localization of the MFS metastasis. MFS's clinical presentation, histological features, and its surgical treatment are also discussed. This work was done in compliance with SCARE checklist .1.1In September 2019, a 70 year-old male patient presented with diplopia in his right eye that progressed during the last two months, in addition to limited right eye movement and right temporal headache. His past medical history included the surgical removal of myxofibrosarcoma tumor from his left gluteal region 5 years ago. It was treated by neo-adjuvant radiotherapy followed by wide local surgical excision and finally by adjuvant radiotherapy. The ophthalmic examination revealed a diplopia and paralysis of abducens nerve in his right eye, normal left eye examination, normal visual acuity and normal appearance of optic disc of both eyes.Head and neck MRI revealed a low signal mass in T1-Weighted image with contrast -A and HiUnder general anesthesia, a skin incision was made on the anterior margin of the sternocleidomastoid muscle at the level of the hyoid bone to expose the right external carotid artery. It was ligated superiorly to the branching point of the superior thyroid artery to avoid excessive bleeding. A Weber-Fergusson incision was made from the middle of the upper lip, along the nasofacial groove to the medial canthus, and the incision was extended below the subcilliary line to the lateral canthus. The skin and musculature layers in the area were swung laterally to expose and ligate the infra-orbital neurovascular bundles. The bone flap of the anterior surface of the maxillary sinus was removed separately. The posterior and medial walls of the maxillary sinus along with the perpendicular plate of the palatine bone were swung laterally, and the internal maxillary artery was ligated and cauterized. The tumor in the pterygopalatine fossa was completely isolated and the maxillary nerve (V2) was intact. The tumor was resected with the posterior wall of the maxillary sinus using the standard partial maxillectomy approach. The dissection was carried on until the round foramen and dura mater were reached. An endoscope was used to make sure the entire tumor was resected -A. The rGross examination showed a white soft tissue mass. The microscopic examination revealed moderate pleomorphic spindle cells in variable shapes and sizes in a myxoid stroma with nuclear atypia, scattered mitosis, and a considerable curvilinear reactive vascularity. These findings were compatible with a grade 2 myxofibrosarcoma  – A,B. IThe patient was scheduled for having 30 sessions of adjuvant radiotherapy 5 days a week for 6 weeks. Unfortunately, the patient did not start the treatment on time and did not fully commit to it. So after four months of the surgery, the tumor recurred at the same location of the previous surgery. However, the patient refused to receive any additional treatment. After 6 months of follow up, his general health status was still stable.2Myxofibrosarcoma presenting in the head and neck region is extremely rare. We can conclude that the tumor of this case had metastasized to this area because of the patient's history of having a myxofibrosarcoma 5 years ago, putting him in the window range of having recurrent distant metastasis. According to a recent study, the range of having distant metastatic recurrence of the MFS is 2.5 months–7.7 years .The unique symptomatology that the patient presented with in this case is attributed to the rare localization of the tumor in the pterygopalatine fossa. Whereas in the majority of cases that presented with diplopia manifested as a mass in the mid-cheek or temporomandibular joint areas .MFS has a low potential of giving distant metastasis. When it metastasizes, it is usually via the hematologic route rather than the lymphatic one. The lungs are the most common destination of MFS metastases . MalignaThe complete resection was very challenging due to anatomical complexity of pterygopalatine fossa. Total maxillectomy, combined with anterior craniotomy have been used as a standard procedure for such cases, resulting in significant postoperative masticatory and cosmetic problems . On the Distant metastasis of MFS occurs in 20% of the cases with a third grade tumor and 4% in patients with a second grade tumor . We high3In conclusion, we reported this case to highlight that metastasis to the pterygopalatine fossa should be considered when a patient with myxofibrosarcoma history presents with neuro-opthalmic symptoms and signs.We also recommend partial maxillectomy as a minimally invasive therapy with adjuvant radiotherapy, to resect tumors in the pterygopalatine fossa that do not aggressively invade the maxillary bone and sinus. This procedure preserves the visceral cranial bones and the associated neurovascular bundles.No ethical approval was needed.Written patient consent was obtained before reporting this case.All data are available from the corresponding author on reasonable request.Not commissioned, externally peer reviewed.All the authors declared that they have no conflicts of interest.


Primary pancreatic hydatid disease is extremely rare. Diagnosis of the disease is difficult because hydatid cysts can be confused with a pseudocyst or neoplastic cystic diseases. Authors report a case of a surgically treated hydatid disease of the uncinate process of pancreas. In a 34-year-old patient with minor symptoms, a cystic disease of the pancreas was accidentally identified. CT scan revealed a multivesicular cystic mass with a maximum of 13-cm diameter and with a calcificated wall. During laparotomy, the uncinate process of pancreas was resecated and the cystic lesion was enucleated. Patient was recovered without complications and recurrence of the disease. There must be a suspicion of hydatid disease when cysts are identified in good conditioned, asymptomatic patients, or in case of wall calcification or multivesicular cysts revealed by radiological images. Surgical procedures are recommended in uncertain diagnoses too, because differentiation preoperatively between cystic pancreatic lesions is often impossible. Echinococcus granulosus. The most commonly affected organs by hydatid disease are the liver and the lung. Other organs, e.g., muscles, kidneys, brain, bones, pancreas, and spleen, are rarely involved [Hydatid disease is caused by the larval stage of involved .Primary pancreatic hydatid cysts are very rare. Diagnosis of the disease is difficult because hydatid cysts can be confused with pseudocysts or neoplastic cystic diseases of the pancreas. The incidence of primary pancreas hydatidosis is 0.2%–2% of all hydatid disease in endemic countries –3. The cAuthors report a case of a surgically treated pancreatic hydatid disease, which caused minor abdominal symptoms, and the final diagnosis of the disease was made intraoperatively.Fig. . CT identified the contact between the lesion and the pancreas head. Radiological imaging did not reveal any cystic lesion neither in the liver, nor in other parenchymal organs. At this time, the patient had only abdominal distress.A 34-year-old male patient’s case history was examined with no any of serious diseases. The patient had complained about lower abdominal pain for a year. Ultrasonography (US) revealed a 6 × 4 × 4 cm solitary cystic lesion with clean wall in the pancreas head region. Connection between pancreas and the cyst could not have been clearly identified by US. Tumor marker (CA 19-9) levels were in the normal range. Patient applied for control only 12 months after the first examination. At that time, computed tomography (CT) scan revealed a multivesicular cystic mass with a maximum of 13-cm diameter with a calcificated wall During physical examination, a 10-cm diameter resilient swelling could be palpated in the epigastric region. Liver enzymes, amylase, lipase, and inflammation markers were in normal range. Blood eosinophilia could not been observed. Echinococcus serology test was negative by enzyme-linked immunosorbent assay. There was a possibility of pancreas cyst, pseudocyst, and hydatid disease as well. In regard to the size of the lesion and the uncertain diagnosis, a laparotomy was offered to the patient.Fig. . The cystic lesion was enucleated and the uncinate process of pancreas was resecated Fig. . After an eventless postoperative period, patient was discharged from hospital on the 7th day after the surgical procedure.During the exploration, a cystic mass with different sizes of daughter cysts was found in the uncinate process of pancreas infiltrating the retroperitoneum Pathological examination of the enucleated lesion confirmed the diagnosis of hydatid disease. Patient received albendazole treatment during follow-up. He had no symptoms and complaints 6 months after the operation.In hydatid disease, liver is involved in 60%–70% of all cases, followed by the lungs 10%–25%) and in sporadic cases in other organs [0%–25% an1The patient in our case was also almost asymptomatic. Most frequent complaint is epigastric pain or feeling of repletion. Localization in the pancreas head or large cysts can cause jaundice, portal hypertension, and rarely acute pancreatitis, and rupture of the cysts can cause anaphylactic shock , 7.In the presented case, the serological test was negative that is not unique in the literature . In typiSurgical procedures are primary in the treatment of pancreatic hydatid disease. Hydatid cysts in the pancreas corpus or tail can be treated by pericystectomy, central pancreatectomy, and distal pancreatectomy with or without splenectomy , 7. WhenResection is also recommended when one cannot exclude securely the possibility of neoplasia. An alternative treatment can be the percutan cyst puncture, aspiration, injection of hypertonic saline solution or absolute alcohol, then reaspiration, or placing a pig-tail catheter into the cyst . TreatmeWhen pancreatic fistula develops after the surgical procedure, the fistula can be sutured, but cystogastrostomy or Rouen-Y pancreaticojejunostomy can also be made . BecauseA review that analyzed eight cases of hydatid disease causing pancreatitis does not report recurrence in any patient . AnotherIn conclusion, primary pancreatic hydatid disease is rare. Caution is needed when cysts in the pancreas are identified in good conditioned patients, the disease is asymptomatic, or in case of wall calcification or multivesicular cysts revealed by radiological images. Surgical procedures are recommended in uncertain diagnoses, because differentiation between hydatid cysts, pseudocysts, and neoplastic cysts can often be confirmed only by pathological examination of the resecated cyst wall.


Foramen of Winslow hernia (FWH) is a rare but emergent condition caused by an increase in the foramen diameter, visceral mobility, and intra-abdominal pressure. To the best of our knowledge, this is the first study to report a case of FWH secondary to large uterine fibroids that was successfully treated with laparoscopic surgery.A 52-year-old woman with large uterine fibroids was diagnosed with FWH. Because of the absence of signs of bowel ischemia and peritonitis, we performed an elective laparoscopic surgery through a 5-port system after bowel decompression using a long intestinal tube. Although foramen of Winslow closure was not performed, her postoperative course was uneventful.Laparoscopic surgery for FWH is considered an extremely effective surgical treatment option because of its safety and efficacy in performing delicate procedures (such as adhesiolysis) with a good magnified field of view. The foramen of Winslow is the entryway to the lesser sac located anterior to the inferior vena cava and posterior to the hepatoduodenal ligament . Due to The most common herniated organ is the small intestine 60–70%), followed by the terminal ileum, cecum, and ascending colon (25–30%) . Althoug0–70%, foSince FWH often develops rapidly with signs of bowel strangulation and ischemia, urgent surgical treatment is required. Through increasing technological innovation, laparoscopic surgery has been widely used for FWHs , 9, 10.To the best of our knowledge, no other study has reported a case of FWH caused by uterine fibroids. Here we report a case of FWH secondary to large uterine fibroids that was successfully treated with elective laparoscopic surgery under preoperative bowel decompression using a long intestinal tube.A 52-year-old woman with unremarkable medical and surgical history presented to the emergency department with intermittent abdominal pain. Physical examinations revealed a soft abdomen with epigastric tenderness without signs of peritonitis. A gently uplifting mass from above the pubis to below the umbilicus was palpated. The patient’s white blood cell count was slightly increased to 12,220 cells/μL , and other serum tests  were within respective reference ranges. Abdominal contrast-enhanced CT showed the presence of large uterine fibroids in the pelvis and herniation of dilated small bowel loops between the portal vein and the inferior vena cava  than male cases (n = 2), the incidence of FWH was higher in males than in females [Table ases n = , the inc females . Despite females , 26. In Foramen closure was performed in only two cases Table . The opeWe experienced a rare case of FWH due to large uterine fibroids. Laparoscopic surgery for FWH is considered to be a very effective treatment option not only because of its minimally invasive nature, but also its utility to perform delicate procedures (such as adhesiolysis) with a good magnified field of view. Treatment of the large uterine fibroids should be considered to prevent the recurrence of FWH rather than foramen of Winslow closure.


Histopathological evaluation is the only method to confirm diagnosis.•Usually seen in association of a vital tooth, but in this case it was seen in a non-vital tooth.•LPC is usually seen within bone, but in this case besides Intrabony component there was a gingival presentation as a swelling. Lateral Periodontal Cyst (LPC) is considered as a rare developmental odontogenic cyst. It is often diagnosed as an incidental radiographic finding, presenting as a circumscribed round radiolucent area between the roots of vital teeth. LPC usually does not present any clinical features. Differentiating the origin of the lesion from an endodontic or periodontal perspective presents as clinical challenge.A female patient presented with an asymptomatic gingival swelling in the lingual aspect of mandibular anterior region. The associated tooth (#34) was endodontically treated 3 years back. A periapical radiograph showed a well-defined round radiolucency on the tooth. Cone beam computed tomography (CBCT) revealed extensive bone destruction. The lesion was surgically excised and histological examination confirmed the diagnosis of LPC. The site healed satisfactorily post-operatively. The case was followed up for a year without any recurrence seen.LPC is a very rare clinical entity, the diagnosis of which requires a detailed case history taking, clinical and radiographic examination are essential to get proper assessment of the pathology. It is said to originate from either the remnants of dental lamina, reduced enamel epithelium or rests of Malassez. LPC presents with a typical histological picture which ensures the confirmatory diagnosis. Surgical enucleation with thorough curettage is the treatment of choice.By reporting this rare case, we would like to stress to clinicians that there are a wide range of cysts and anatomic structures are present in the canine-premolar region of mandible of which LPC is a rare possibility. It is a relatively rare odontogenic lesion, accounting for just 0.4% of all odontogenic cysts . LPC hasFrom a clinical point of view, it is important to correctly diagnose LPC from a number of other lesions which present similar clinical or radiographic picture. The list includes gingival cyst, odontogenic keratocyst, lateral radicular cyst, glandular odontogenic cysts, lesions of endodontic and periodontal origin ,9,12. A We present a case of LPC which located in the canine-premolar region of mandible which occurred in an endodontically treated tooth which has not been reported often. The use of Cone beam computed tomography (CBCT) to assist in the diagnosis and treatment planning is also discussed. A brief literature review of the clinical, radiographic and histological features of LPC is also presented.This case has been reported in accordance to the SCARE criteria .2A 49 year-old female patient reported to a private practice at Trivandrum, with a complaint of painless swelling in the lingual aspect of left mandibular canine [#33] and first premolar [#34] region of three months duration. The patient did endodontic treatment of #34 for deep caries three years previously and a crown had been placed. The patient reported no systemic comorbidities, was in good general health.Upon clinical examination, a well-circumscribed and fluctuant swelling, measuring about 10 mm in diameter, was observed at the junction of the buccal attached and free gingival margins in between the teeth #33 and #34 A. The swAfter getting informed consent from the patient, scaling and root planing was done. Based on the clinical and radiographic findings, a provisional diagnosis of LPC was given. A gingivectomy procedure was done in the region. The enlargement was completely removed and curettage of the area was done. The bone defect showed expansion of the lingual cortical plate with a smoothened appearance of the bone A. SutureThe excised soft tissue specimen was sent for histopathology examination (HPE). HPE of the excised specimen showed cystic lumen lined by 2–3 layers of non-keratinizing cuboidal cells resembling reduced enamel epithelium which were hyperplastic at places. The lining epithelium also showed clear cells, localized thickenings/plaques and mural bulges protruding into the cystic cavity. The underlying connective stroma is dense collagenous made up of bundles of collagen fibres, fibroblasts, blood vessels and dense infiltration of chronic inflammatory cell infiltrate. The overlying epithelium is parakeratinized stratified squamous in nature and is separated from the lesional tissue by a zone of normal connective tissue stroma A–D. Base3It was 1958, that Standish and Shafer first reported LPC in literature with a series of five cases presenting in the mandible . LPC is In the etiopathogenesis of LPC, there are mainly three different views with regard to cell type of origin. Altini M and Shear M have attributed origin from reduced enamel epithelium (REE), due to the fact that the cyst is often lined by nonkeratinized epithelium similar to REE as has been shown by PCNA immunohistochemical expression . Cohen DLPC has a characteristic radiological presentation as a round or oval shaped well circumscribed radiolucency with a sclerotic border located between the crest and apex of alveolar process. It does not involve periodontal ligament space or cause root resorption of adjacent teeth. A similar radiographic picture may be seen in anatomic interradicular radiolucencies such as the mental foramen, nutrient canals and the maxillary sinus or in pathologies such as cysts of pulpal origin, odontomas, tumours and other cysts of jaw . In 2006Based on the clinical and radiographic findings, a provisional diagnosis of LPC was considered and HPE was done to confirm this. LPC presents some characteristic histopathological features which include one to three cell layer thick non-keratinized epithelial lining which often resembles odontogenic epithelium or REE, which may display a palisading pattern. The epithelial lining shows areas of thickening referred to as epithelial plaques which are composed of glycogen rich clear fusiform cells. The connective tissue is rich in collagen, which may show areas of hyalinization with minimal inflammatory cell infiltrate ,16. In tBotryoid odontogenic cyst (BOC) also reports similar HPE findings as LPC. The differentiating feature is that BOC is seen as a multilocular radiolucency. Some authors consider BOC as a histopathologic variant of LPC, which has a greater tendency for recurrence following treatment. Altini and Shear in 1992 proposed a new classification of LPC into unicystic, polycystic or botryoid variants, all of which have a similar histopathological presentation. This classification was based on the hypothesis that all LPCs can progress to multicystic lesions . A diffeLiterature shows that in most of the cases of LPC, the associated teeth are vital therefore extraction or endodontic treatment is not required. Treatment of LPC is conservative surgical enucleation and thorough curettage of cystic lining to remove any remnants ,13. Guid4LPC is a rare developmental odontogenic cyst which should be considered in the differential diagnosis if in a radiograph cystic lesion is seen on the lateral surface of tooth especially in the mandibular canine-premolar and anterior maxillary region. It may present as an asymptomatic gingival swelling which needs to be thoroughly examined both clinically and radiologically. A histological evaluation is necessary to confirm the diagnosis. The treatment of choice is surgical removal and recurrences are infrequent.The authors report no declarations of interest.There is no funding from any source for this paper.Written informed consent has been obtained from the patient. Her approval for publication of case details has also been received.Since the case has been treated in a private clinic. Permission has been obtained from the clinic authorities for publication of case details.A fully informed written consent has been obtained from the patient.Roshni Ramesh: Conceptualization, methodology, validation, investigation, resources, writing-revision & editing & supervision.Arun Sadasivan: Conceptualization, methodology, validation, investigation, resources, writing- original draft, visualisation & supervision.N/A.Dr Arun Sadasivan.Not commissioned, externally peer-reviewed.


The importance of this paper is to help to emphasize the importance of chemotherapy for children with pure intratesticular rhabdomyosarcoma after radical inguinal orchiectomy is done as first treatment of rhabdomyosarcoma. The information provided in this paper about the follow-up outcomes of the patient described in this paper, it highlights that, recurrence and even metastasis of intratesticular rhabdomyosarcoma in children are more likely to occur if surgery it not combined with chemotherapy.Herein, we present a 6-year old African male child with a 3 months history of a painless right intratesticular tumour. The tumour was poorly vascularized and was in continuity with the spermatic cord. Pelvic computer tomography (CT) scan showed a heterogeneous mass with well-defined margins without microcalcification and multiple bilateral inguinal enlarged lymph nodes were noticed without pelvic lymphadenopathy. The tumour measured 3.8 × 2.8 × 3.9 cm. The tumour marker panel showed: lactate dehydrogenase of (472 UI/l), alpha-fetoprotein (1.43 UI/ml) and human chorionic gonadotrophin beta (2.9 mUI/ml). Microscopically, the tumour was composed of small to medium size undifferentiated cells. These were oval to spindle, hyperchromatic cells to stromal myxoid degeneration were noted. Tunica albuginea and rete testis both were infiltrated by tumour. The tumour showed high mitotic count which measured 50 mitoses per 10 High Power Field (HPF). The diagnosis of rhabdomyosarcoma (RMS) was confirmed by immunohistochemistry (IHC) testing using myoD antibody which showed strong and diffuse intranuclear staining of the tumour cells. Currently, he is on cyclophosphamide and vincristine chemotherapy regime and his condition has improved much.The experience obtained from the index case is crucial for the management of patients with intratesticular rhabdomyosarcoma which should always make sure that radical inguinal orchiectomy is covered by chemotherapy and/or radiotherapy. This will potentially lower the possibilities of recurrence and/or metastasis of the tumour, hence improving the prognosis of the patients. We report the clinical, radiological, and laboratory characteristics as well as the outcome of the patient. Rhabdomyosarcoma (RMS) is a common mesenchymal malignancy in the pediatric population with a slight male preponderance , 2. In 2Primary intratesticular rhabdomyosarcoma (PITRMS) is a rare intrascrotal tumour localized in the tunica vaginalis, epididymis, or spermatic cord. Diagnosis of PITRMS can be done on a high degree of clinical suspicion with the help of imaging diagnostic tests, biopsy and immunohistochemistry , 7. PITRHerein, we report the case of a 6-year old African male child who was diagnosed with pure intratesticular RMS of the embryonal variant. This is extremely rare with an incidence of only 1% . We deciA 6-year old African male was admitted in the pediatric ward with a 3-month history of a right testicular mass which was painless. He had no history of trauma, difficulty in passing urine or blood in urine. The parents denied any history of cancer in the family. His past medical, social, family and environmental history were uneventful. Either it was reported that, the patient received Diphtheria, Tetanus, Pertussis and Haemophilus influenza and Hepatitis B, pneumo-conj, and Measle-Rubella (MR) vaccines at the right schedules. On admission, physically, the patient was ill-looking, pale and febrile (39.2 °C). His weight and height were 20.2 kg and 86 cm, respectively with pulse rate and blood pressure of 68 beats per minute and 127/73 mmHg, respectively. The right testis was swollen, painless, and non-tender, nodular and firm in consistency. The overlying scrotal skin was shining with engorged overlying blood vessels. His neurological examination revealed normal motor function , normal sensory function and normal reflex responses.The laboratory tests included the following: haemoglobin level 8.3 gm/dl (low), aspartate transaminase (AST) 10-36 IU/l , alanine transaminase (ALT) 36-39.1 IU/l , serum creatinine 0.81 mg/dl , protein in urine 3.1 gm/ml , human immunodeficiency virus (HIV) and hepatitis B surface antigen (HBsAg) were both negative. Pelvic computed tomography (CT) scan showed a heterogeneous mass with well-defined margins without microcalcification , alpha-fetoprotein (1.43 UI/ml) and human chorionic gonadotrophin beta (2.9 mUI/ml). The patient’s tumour was staged as clinical stage I. By using the criteria for risk stratification of patients with RMS established by the Children’s Oncology Group for Soft-Tissue Sarcoma (COG-STS) committee  which adThe pelvic, abdominal and chest CT scans showed no metastases to either lungs or mediastinum. The patient had operation (orchidectomy). During the operation, external oblique aponeurosis was opened; ilio-inguinal nerve was isolated and secured. Inguinal canal contents were dissected. There was no vas deferens that was seen and the left testis was infiltrated by the tumour and the tumour had gone beyond the tunica vaginalis. A straw colored jelly-like material was seen surrounding the tumour within the scrotal sac.The dissected inguinal canal contents were clamped, ligated with vicryl 3/0 and transected. The malformed testis by the infiltrating mass was dissected and excised and hemostasis was achieved. The specimen was taken for histological evaluation. Retroperitoneal lymph node dissection was not performed in our case as radiologically there was no lymphadenopathy. Macroscopically, the submitted specimen comprised of a grayish-yellow fibro-fatty tissue that measured 4 × 3 × 2 cm. The cut surface showed a lobulated white-tan and firm mass  had been done because the patient delayed to start chemotherapy on time until when the parents saw a mass which was gradually increasing in size. Then he was taken back to hospital where he was given chemotherapy after evaluation with chest CT scan to rule out for metastasis which revealed normal findings. The chemotherapy agents used were 1.2 mg iv cyclophosphamide, 0.94 mg iv actinomycin D and 1032 mg iv vincristine which he completed after 5 months. We followed-up the patient for one year and his postchemotherapy period was uneventful. Currently, the child is healthy and he is continuing with schooling.This paper presents the case of a 6-year old African male with primary intratesticular RMS of embryonal variant which was localized. However, it recurred following radical inguinal orchiectomy after delaying to start chemotherapy. This case is unique in the sense that the patient is relatively younger compared to majority of the cases reported in the English literature. The other unique feature of this patient is that the patient falls in the low risk group based on the risk stratification of RMS.The histogenesis of PITRMS is still enigmatic. It has been theorized that, either PITRMS originates from dedifferentiated mesenchyme having the capacity for rhabdomyoblastic differentiation, or perhaps from embryonal muscle tissue that has been displaced during the early stages of tissue development, but not from metaplasia of the connective tissue or smooth muscle . AnotherIntrascrotal RMS can either be intratesticular or paratesticular. PITRMS are very rare and they comprise less than 1% of all the testicular sarcomas whose prevalence is 2% of all testicular tumours. On the other hand, studies have reported that, paratesticular RMS have an incidence of approximately 7% of the intrascrotal RMS . PrimaryPITRMS clinically presents as painless intrascrotal masses which may progress for a few weeks and become painful and a few cases have been reported to present with pain. Similarly, trauma is associated in only 7% of cases and is not thought to be causative but rather to draw attention to the tumour . UsuallyDiagnosis of PITRMS is based on different approaches. This includes imaging , IHC staining, gross appearance and histopathology. Scrotal ultrasound and abdominal CT scan may be used to determine an intratesticular or paratesticular origin of the scrotal mass . UltrasoTreatment of PITRMS still is not comprehensive and trials have not been explorative due to limited availability of cases because of the rarity nature of the disease. A multidisciplinary therapeutic approach which involves surgery and chemotherapy with or without radiotherapy has been established based on risk stratified groups . RadicalFrom the observations in the IRS-III, three recommendations were put forward which include (1) Treatment for children with RMS should be based entirely on risk stratification, site of the primary tumour site and the extent of disease (2) Children with low risk groups (group I or II) tend to respond well to RIO and chemotherapy (3) Radiotherapy should be provided for children with risk group II only . This trRetroperitoneal node clearance is controversial and is probably not justified for staging or in initial treatment, but it has a role in debulking disease if nodes persist after chemotherapy . There iPrognosis of PITRMS is said to be poor due to its aggressiveness nature of the tumour. A number of prognostic factors determine the clinical outcomes of any patient with rhabdomyosarcoma including those with PITRMS. Age of the patient at diagnosis, histological type, retroperitoneal lymph node involvement (RPLI), risk group, site of the primary tumour, intensification of treatment and metastasis altogether play a major role in prognostication of the disease for children, adolescents and adults , 11, 22.et al. reported that tumour-cell hyperdiploidy correlates with a favorable outcome in children and young adults with embryonal RMS [et al. reported that t characterizes alveolar RMS with a poorer prognosis [t breakpoints has been reported to identify a fusion gene, PAX3-ALV, that can now be readily identified by the reverse polymerase chain reaction [The use of molecular tests in complementing the process of determining the prognosis of patients with RMS seems to provide promising results. For example, Pappo onal RMS . Also Dorognosis . Moleculreaction . Lack ofFrom the observation of the case presented in this paper, it may be learned that, radical inguinal orchiectomy should always be followed immediately with adjuvant chemotherapy in order to prevent recurrence of the resected primary tumour as it was the experience in our patient. Also the use of ancillary tests such as molecular prognostic factors would be of paramount importance in improving the clinical outcomes of children as well as adults diagnosed with PITRMS.


A fecaloma is a mass of accumulated feces with a consistency much harder than that of a fecal impaction. It is most frequently observed in the rectum and sigmoid area, and associated complications include colonic obstruction, ulceration, bleeding, and perforation. A one-year-old, previously healthy boy with no history of chronic constipation was admitted because of vomiting and abdominal distension. An abdominal computed tomography scan showed small and large bowel distension due to multiple obstructive fecalomas in the transverse colon. As the fecalomas could not be resolved by laxatives, enemas, or colonic lavage, endoscopic disimpaction under general anesthesia was attempted. Repeatedly shaving the fecalomas with biopsy forceps finally resulted in gradual fragmentation with subsequent passage. Gastrointestinal food allergy was later suggested as the cause because eosinophilic infiltration was found in a biopsy specimen of the colon wall. Endoscopic disimpaction is an effective treatment approach for addressing fecalomas to avoid more invasive surgical intervention. A fecaloma is a laminated mass of accumulated feces and has often been reported in patients suffering from chronic constipation –11. The A one-year-old, previously healthy boy with no history of chronic constipation presented with a five-day history of frequent vomiting, constipation, and abdominal distension. He was 74 cm in height and weighed 9 kg at the time of presentation. Upon examination, he was tachycardic but otherwise relatively stable. His abdomen was considerably distended but nontender and filled with palpable stool, yet a rectal exam revealed no stool. An abdominal X-ray scan demonstrated multifocal air-fluid levels . SuspectA colonoscopy was performed using a scope of the upper gastrointestinal tract  with insufflation of carbon dioxide. Edematous mucosa of the descending colon and multiple giant brown fecalomas were observed to be occupying the lumen of the transverse colon . AlthougAlthough fecal impaction is a common and disturbing problem, a fecaloma is a particularly rare form of impaction in which a mass separable from the rest of the bowel contents is formed. Fecalomas are most frequently seen in the rectosigmoid area because the stool becomes firmer, and the colon diameter is smaller in this area , 9. On tRegarding the treatment of fecalomas in the rectosigmoid area, conservative procedures such as bowel rest, laxatives, enemas, manual evacuation, and colonic lavage are commonly adopted –3. HowevWhile the majority of published cases occurred in adults, fecalomas sometimes appear in pediatric patients; however, there are no reports in which the fecalomas were dissolved by endoscopy , 8, 11. γ and tumor necrosis factor-β induce edema of the gastrointestinal mucosa, which causes intestinal peristalsis depression [There are multiple causes of fecaloma formation, but in our case, the cause was not immediately evident as the patient had no reported history of altered bowel habits. Chronic constipation is a common problem that can lead to fecal impaction and even the development of fecalomas –4. Otherpression . In our In conclusion, we herein report a case of fecalomas inducing small and large bowel obstructions in an infant due to a suspected gastrointestinal food allergy. The endoscopic disimpaction of fecalomas is arduous and requires a great deal of time to perform but is an ideal treatment for use in patients without an imminent condition to avoid the need to progress to surgical intervention.


Ameloblastic carcinoma (AC) is a scarce malignant tumor which is more prevalent in the mandible than the maxilla. It occurs in a wide range of age groups, and there is a sex predilection in males. AC shows specific microscopic features and requires more aggressive surgical treatment plans in comparison with conventional ameloblastoma. Radiographically, AC resembles ameloblastoma except that it rarely represents focal mineralized materials, seemingly reflecting dystrophic calcification. This characteristic is uncommon in typical ameloblastomas, and only few cases reported with such opacities and mineralized materials. Due to this rare radiographic and microscopic presentation, an accurate diagnosis could be challenging, and pathologists should consider a combination of benign and malignant odontogenic tumors occurring in jaws. Odontogenic malignancies are rare lesions which comprise one percent of all cysts and tumors happening in the jaws arising from tissues associated with odontogenic epithelium –3. AC isConsidering the rarity and diverse nomenclature of this lesion, AC has been a topic of diagnostic challenges for a long period of time . The mosRadiographically, AC looks like ameloblastoma except that it occasionally shows focal radiopaque materials; apparently, it reflects dystrophic calcification itself. This trend does not often appear in typical ameloblastomas . The litWe documented a case of recurrent AC with calcification in a 40-year-old woman who underwent surgery and radiotherapy.A 40-year-old woman referred to Imam Khomeini Cancer Institute was complaining of intense lower facial pain and progressive swelling for the period of the last six months. Although she has not had a considerable problem in her past medical history, she has also given us a history of her left second premolar and first molar teeth root canal therapy in which she had not noticed any change in pain and swollen area size. She had also undergone incisional biopsy in another health care center with pathologic results designated as a “malignant Pindborg tumor.”On clinical examination, diffuse swelling causing lower lip tenderness and paresthesia with extension to the left posterior part of the mandible was detected. On examination, a mass that measures approximately 4 cm with a rubbery consistency was found. The overlying skin tissue was intact; however, oral mucosa was ulcerated. No palpable subcutaneous cervical lymph node was found. There was no difficulty in swallowing, dysphagia, trismus, or dysphonia.On panoramic and cone-beam computed tomography (CBCT), the lesion with a well-defined border and internal focal radiopacities was seen that extended from the periapical area of the mandibular left canine to the mandibular left second molar in an anteroposterior dimension and from the alveolar crest to the inferior border of the mandible in a superoinferior aspect . Buccal Microscopic examinations showed odontogenic epithelial neoplasm composed of sheet and island of epithelial cells with malignant features including cellular pleomorphism, hyperchromatism, few atypical mitotic figures, central necrosis, and perineural invasion in a collagenized stroma. At the periphery of the islands, tall columnar cells resembling ameloblast cells with nuclear palisading, reverse polarity, and subnuclear vacuolization are evident. The particular feature of this tumor was the presence of intercellular eosinophilic amorphous materials in extensive areas that had been transformed into mineralized materials in some parts. The lesion was superficially covered by ulcerated stratified squamous epithelium . BesidesOdontogenic malignancies are rare lesions which are composed of one percent of all cysts and tumors appearing in the jaws emanating from tissue associated with odontogenic epithelium , 5, 7, 8Varying signaling pathways which take part in the progression of ameloblastomas have been recognized highlighting the MAPK signaling pathway with its predominant B-RAF proto-oncogene serine/threonine kinase (BRAF) V600E mutation . Since tBased on a systematic literature review which has been carried out by Deng et al. from January 2000 to July 2018 and our literature review, AC presents a higher male prevalence when compared to females. The most involved site in the jaw is the posterior area of the mandible with a 64% occurrence rate . AC can Based on radiographic view, differential diagnosis of posterior mandibular radiolucency includes odontogenic keratocyst, myxoma, Pindborg tumor, ameloblastoma, and less commonly intraosseous malignancies or metastatic lesions , 11. TheDifferentiation between ameloblastic carcinoma and classic ameloblastoma may be impossible based on the radiological aspect solely. Both of the lesions demonstrate a bony hard expansive mass with teeth displacement, loss of lamina dura, root resorption, and occasionally teeth loosening , 13. LikAs mentioned in the radiographic features of the discussed case, there were severe buccal and lingual cortical plate perforation, root blunting of a mandibular first premolar, and root resorption in a mandibular first molar as well as the appearance of dystrophic calcifications. The notable radiologic feature of this patient was the presence of multiple small internal radiopacities throughout the lesion. Based on our literature review, two cases of AC have been represented with calcification , 16. SimOn occasion, calcification may be a result of intensive apoptotic activity, and the final differentiation of keratinocytes is taught out to be a variant of the apoptotic process , 17, 18.Looking at the microscopic features of this case in a different angle reveals that the tangible absence of stellate reticulum-like cells in the center of islands, trabeculae, and sheets of the neoplastic cells is highly in favor of AC. In this regard, Angiero et al. pointed out that the presence of nests, islands, sheets, or trabeculae of reverse polarized epithelium with scarce or no existence of stellate reticulum-like areas can be considered a diagnostic clue for the pathologist to appraise AC in differential diagnosis . The Ki-There are different methods to tackle AC, including surgical resection with a safe margin with or without cervical lymph node removal and surgical removal with or without chemotherapy. Having a safe margin can reduce the rate of recurrence to less than 15% , 27. AnoOur case underwent hemimandibulectomy and radiotherapy. We followed the patient for four years after the initial diagnosis of AC. She is still alive but unfortunately suffering from multiple lung metastases and poor general health quality.Dystrophic calcifications are rare findings in ACs. However, the presence of such materials as an occasional feature can be considered a diagnostic point for pathologists. It seems that more cases are needed to be studied to understand how these calcifications could relate to the patient's outcomes.