Esophageal cancer including squamous cell carcinoma (SCC) and adenocarcinoma represents 4% of all cancers in the United States. Patients with esophageal cancer frequently present with locally advanced disease, and about 40% of patients have evidence of metastatic disease on presentation. Common sites of metastasis include liver, lung and bone. Here, we present a rare case of colonic metastasis from primary esophageal SCC.A 60-year-old Caucasian male with a history of 20-pack-year cigarette smoking received surgery and adjuvant chemoradiotherapy for locally advanced SCC of larynx. Approximately 9 months later, he developed dysphagia, and found to have a esophageal SCC in the mid-esophagus with regional lymph node involvement. He underwent chemoradiation treatment with good response and improved symptoms but declined subsequent surgical resection for esophageal cancer. About 1 year after the diagnosis of esophageal cancer, he developed blood streaked bowel movement and severe anemia. Colonoscopy showed a 3-cm mass in the proximal ascending colon; biopsy showed metastatic SCC, consistent with metastasis from esophageal primary. He subsequently received palliative radiation to the ascending colon metastatic tumor with improvement of anemia, and remained transfusion independent for more than 3 months.Colonic metastasis from esophageal SCC is rare, and associated with poor prognosis. There are no definite features in terms of location, histological differentiation etc. that contribute to colonic metastasis from primary esophageal SCC. The goal of treatment is palliative and data from our and other case reports support the use of chemotherapy and radiation for symptom improvement and disease control. Esophageal cancer including squamous cell carcinoma (SCC) and adenocarcinoma represents 4% of all cancers in the United States; in 2016, it is estimated approximately 16,000 new cases and more than 15,000 deaths as a result of this aggressive disease . PatientIn July 2009, a 60-year-old Caucasian male with hypertension and dyslipidemia presented with neck stiffness and right neck mass for 3 months. The patient had a 20-pack year history of smoking and moderate alcohol intake. Initial ENT evaluation with direct laryngoscopy showed some fullness in the right base of the tongue without any abnormality at the larynx or pyriform fossa. CT scan of neck demonstrated a 3.8-cm necrotic mass in the right neck. Ultrasound guided biopsy of the right neck mass was consistent with malignant cells favoring poorly differentiated SCC. Patient was referred to our center for further work up. Repeat direct laryngoscopy showed a suspicious lesion at the right aryepiglottic fold. He underwent CT scan of chest, abdomen and pelvis plus PET-CT, which showed a right neck mass (4.2 cm × 3.5 cm) medial to the sternocleidomastoid muscle. A diagnosis of metastatic SCC of the right cervical lymph node with likely primary lesion at the base of the tongue or larynx was made.2 laser laryngectomy and right modified radical neck dissection in September 2009. Pathology from the right supra-glottis laryngectomy showed 0.6 cm tumor, which was consistent with moderately to poorly differentiated SCC with clear margins. Right neck dissection showed 3 out of 21 lymph nodes positive for metastatic SCC with the largest being 3 cm in greatest dimension. There was an extra-nodal extension of the tumor. The final diagnosis was T1N2bM0 (Stage IVA) right supra-glottis laryngeal SCC. Patient received adjuvant treatment with chemoradiotherapy completed in January 2010. Two months after completion of adjuvant treatment, the fiber-optic laryngoscopy and CT scan of neck showed no evidence of disease.Patient underwent right supra-glottis COIn October 2010, patient developed dysphagia, and examination of esophagogastroduodenoscopy and showed a stricture at 31 cm, a friable mass at 31–34 cm, Barrett’s mucosa noted from 34 to 39 cm, and the diaphragm at 39 cm from the incisors, respectively. Endoscopic ultrasound was done but the scope could not be passed through the stricture, but showed a mass at about 24 cm from the incisors in the paraesophageal area just below the aortic arch. Fine needle aspiration biopsy of the esophageal stricture and mediastinal mass showed poorly differentiated SCC, positive for p63 and CK 5/6 by immunohistochemical study. Imaging study with PET-CT and CT scan of neck, chest, abdomen and pelvis confirmed locally advanced esophageal cancer with mediastinal lymph node involvement. He underwent chemoradiotherapy with weekly paclitaxel and carboplatin, completed in February 2011. A follow-up CT scan of chest in June 2011 indicated a good treatment response with tumor shrinkage. He declined surgery after completion of chemoradiotherapy.In November 2011, he developed blood streaked bowel movement and severe anemia with hemoglobin of 5.9 g/dL. He underwent CT scan of abdomen and pelvis and colonoscopy, and was found to have a 3-cm mass in the proximal ascending colon , esophageal SCC. It has been shown that patients surviving laryngeal cancer treatment have increased risk of SPT, usually in the aerodigestive tract; the cumulative risk of developing a SPT is found to be 26% at 10 years and 47% at 20 years . Field cEsophageal cancer is among the top 10 leading cause of cancer-related mortality and common cancer worldwide. There is a decreasing incidence of esophageal SCC but increasing incidence of esophageal adenocarcinoma in the Western world. Most patients present with locally advanced disease which requires combined-modality treatment including chemoradiotherapy with or without surgery. Neoadjuvant chemoradiation followed by surgery has been shown to improve local control, disease-free survival and overall survival compared to surgery alone . For patOur patient declined surgery after chemoradiotherapy for locally advanced esophageal SCC, and developed metastatic disease to ascending colon with severe hematochezia and anemia about 9 months afterwards. His symptoms improved after palliative radiation to colonic metastasis, and remained transfusion independent for more than 3 months. He also developed brain metastasis and received brain radiotherapy before succumbing to esophageal cancer about 6 months after being diagnosed with metastatic disease.The predominant histology type of primary colorectal cancer is adenocarcinoma; less common ones include neuroendocrine tumor, lymphoma, sarcoma and SCC . The incFor patients with metastatic esophageal SCC, the goal of treatment is palliative to improve quality of life and survival, which requires multi-disciplinary approach including pain and symptom management. Our patient declined chemotherapy but received colonic radiation with improved symptoms and independency of blood transfusion lasting for 3 months. Palliative chemotherapy has yet to demonstrate survival improvement compared to best supportive care, but is frequently used with platinum-based regimen . TargeteColonic metastasis from esophageal SCC is rare, and associated with poor prognosis. There are no definite features in terms of location, histological differentiation etc. that contribute to colonic metastasis from primary esophageal SCC. The goal of treatment is palliative and data from our and other case reports support the use of chemotherapy and radiation for symptom improvement and disease control.


The occurrence of extranodal primary B cell non-Hodgkin's lymphoma is rare. Total hip replacement is one of the most common orthopaedic procedures performed. There has been an increased incidence of primary lymphomas involving periprosthetic sites. Chronic inflammation due to metal debris arising from the prosthetic implants has been evidenced as one of the causes for the development of soft tissue lymphomas albeit rarely. We describe a case report of a 77-year-old patient who had underwent a cemented total hip replacement in the past who further developed large B cell primary non-Hodgkin's lymphoma. She presented initially with signs and symptoms highly suggestive of underlying periprosthetic infection. The radiological imaging was also indicative of periprosthetic infection. The diagnosis was eventually confirmed after an open biopsy. This case underlines the importance of considering and including soft tissue malignancy in the differential diagnosis of suspected chronic periprosthetic infection. Primary non-Hodgkin's lymphoma (NHL) is a rare malignancy of the musculoskeletal system and is known to affect up to 5–25% of patients , 2. WithWe describe a unique and rare case report of a patient who presented with signs and symptoms suggestive of periprosthetic joint infection after total hip replacement, which finally concluded to have been diffuse B cell non-Hodgkin's lymphoma.A 77-year-old woman presented to our Accident and Emergency department with a one-year history of a painful right hip replacement. She complained of difficulty in weight bearing and a gradually diminishing walking distance such that in the four weeks prior to admission she was essentially housebound. Furthermore she reported her hip pain had progressed to an extent that it was present at rest and on occasion woke her up from sleep at night.She had been reviewed six months previously by her local GP who had started her on high dose oral steroids for a provisional diagnosis of polymyalgia rheumatic under the guidance of the rheumatologist. There was no history of trauma.Her past medical history included osteoarthritis, iron deficiency anemia, and a right cemented Exeter total hip replacement performed three years previously.On examination she appeared generally unwell and frail. She was apyrexial with no evidence of any localized features of erythema, swelling, redness, or skin changes around the affected hip. There was severe tenderness on palpation around the greater trochanter. Her active range of motion of the right hip was limited to 20° flexion with all other movements being severely restricted. Passive range of motion was also limited.99 and SPECT (Single Photon Emission Computed Tomography) showed a large an area of intense hypervascularity at the medial aspect of the right proximal femur  is a malignant proliferation of either B or T cell lymphocytes in a disorderly fashion. Diffuse large B cell lymphoma is the most common of all lymphomas accounting for about 35% . The disAny part of the musculoskeletal system can be affected in lymphomas and can present as a soft tissue mass resembling either infection or soft tissue tumour , 17. TheThere are several studies which looked at increased incidence of lymphoma after total hip replacement. Gillespie et al. concluded that in the first decade following total hip replacement the incidence of tumours of the lymphatic and haemopoietic systems was significantly greater and this may be as the resultant effect of the prosthetic implants . AnotherUsually diffuse B cell NHL affects patients above the age of 60 years and predominantly occurs in males with a ratio of 1.5 : 1 , 21. TheOne of the differential diagnoses of mass lesions after total hip replacement is a metal-on-metal pseudotumour, also known as aseptic lymphocyte-dominant vasculitis-associated lesion (ALVAL), especially around a metal-on-metal hip or knee replacement . They arThe other cause of psuedotumours after THR is avulsion of abductor tendons associated with fluid collection. MRI scans are diagnostic with pure fluid signals and the anatomical location of the pseudotumour .The treatment of diffuse B cell NHL is combined chemotherapy and radiotherapy with good outcomes . The fivPrimary diffuse large B cell NHL is a rare musculoskeletal malignancy. It can present atypically as a periprosthetic joint infection; therefore clinicians should have a high index of clinical suspicion to consider and include tumour pathology in the differential diagnosis.In cases of suspicion of alternate pathology one should consider an open biopsy for a timely diagnosis and treatment.


Gastric adenocarcinoma of fundic gland mucosa type (GA-FGM) is a rare tumor composed of atypical cells with differentiation toward the fundic gland as well as the foveolar epithelium. Including our case, only 9 cases of GA-FGMs were reported from 2010 to 2016.An 87-year-old man was referred to our institution for endoscopic resection of a gastric lesion. The tumor was classified as type 0-I + IIa according to the Paris classification. Magnifying endoscopy with narrow band imaging (ME-NBI) revealed different structures of crypts and vessels among the components, illustrating the collision of 2 types of gastric cancer.We performed endoscopic submucosal dissection and successfully removed the tumor en bloc.The histological findings differed markedly between the 0-I lesion and the 0-IIa lesion. The superficial part of the 0-I lesion consisted of a papillary structure, and the deeper part consisted of a tubular structure that showed inverted downward growth to the submucosal layer with the lamina muscularis mucosae. Immunohistochemically, the superficial part of the 0-I lesion was positive for MUC5AC, which had differentiated to foveolar epithelium. The deeper part was positive for pepsinogen-I and MUC6, which had differentiated to fundic gland. The 0-I lesion was diagnosed as gastric phenotype of adenocarcinoma differentiated to fundic gland mucosa with upward growth in the superficial part and downward growth in the deeper part. The 0-IIa lesion was composed of a tubular structure positive for MUC2, and it was diagnosed as an intestinal phenotype of well differentiated adenocarcinoma. The boundary was clear, and no transitional tissue was observed between the 0-I and 0-IIa lesions, suggesting that the 0-I + IIa lesion was a gastric collision tumor of GA-FGM and well differentiated adenocarcinoma.We herein report the first case of inverted GA-FGM colliding with well differentiated adenocarcinoma. ME-NBI can be used to diagnose GA-FGM even if the lesion collides with other types of adenocarcinoma. It has been reported that GA-FG accounts for 0.98% to 1.6% of gastric cancer cases,,4 and the number of reported cases has been increasing.,6 Recently, Tanabe et al reported that gastric adenocarcinoma of fundic gland mucosa type (GA-FGM) included a phenotype, showing atypical cells with differentiation toward the fundic gland as well as the foveolar epithelium. However, the endoscopic features of GA-FGM remain unclear, particularly for inverted lesions. We herein report the first case of inverted GA-FGM colliding with well differentiated adenocarcinoma.Gastric adenocarcinoma of fundic gland type (GA-FG) was proposed as a new, rare variant of gastric adenocarcinoma in 2010.2Helicobacter pylori (Hp) by the urea breath test, and he had never received Hp eradication therapy. An endoscopic examination revealed a reddish, elevated lesion with faint reddish area on the great curvature of the gastric lower body  on white-light endoscopy. The area of the inverted GA-FGM showed a sessile lesion (0-I), suggesting that inverted GA-FGM exhibits elevation due to the tumor volume on the deeper side. ME-NBI revealed different structures of crypts and vessels among the components, illustrating the collision of 2 types of gastric cancer. ME-NBI also detected irregularly circular marginal crypt epithelium with irregular vessels within the circular intervening part at the 0-I area, which histologically corresponds to a papillary structure mimicking foveolar epithelium at the GA-FGM part, suggesting that ME-NBI can be used to diagnose GA-FGM even if the lesion collides with other types of adenocarcinoma.,8 , 2 cases were predominantly positive for MUC6 (mucous neck cell dominant type), and 4 lesions were equally stained for Pep and MUC6 (chief mucous neck combination type). Although GA-FG has been reported to be mainly chief cell-dominant type, GA-FGM appears to include various types of fundic gland differentiation.Including the present case, only 9 cases of GA-FGMs were reported from 2010 to 2016–12 The accumulation of similar cases will help clarify the etiology of the inverted growth of gastric cancers.The present case exhibited inverted growth that was thought to be due to the downward growth of GA-FGM. Concerning the inverted growth of gastric lesions, such as gastritis cystica profunda, heterotopic mucosa-related carcinoma, and gastric inverted hyperplastic polyp, injury of the muscularis mucosae caused by chronic gastritis and erosion is thought to lead to inversion into the submucosal layer.No other person contributed to the manuscript.


Staphylococcus aureus bacteremia and back-pain. The patient was suspected of having spondylitis and treated as such with antibiotics; however, both fluorine-18 fluoro-2-deoxy-d-glucose (18F-FDG) positron-emission tomography and magnetic resonance imaging (MRI) with iv contrast showed significant inflammation of muscles and subcutaneous soft tissue in relation to the patients back and left shoulder, but no signs of the working diagnosis of spondylitis. The unusual location of the infection was not explained until a few days prior to being discharged when the patient reported visits to a local physiotherapist where he would have acupuncture performed for non-specific back pain. His last acupunctural procedure had been performed 6 days prior to admission. This case is, to our knowledge, the first to show muscular inflammation on both 18-F-FDG PET/CT and MRI following acupuncture due to S. aureus. This case highlights the need for clinicians to search for alternative explanations when imaging does not support the diagnosis.We describe the clinical course of a 60-year old male admitted with


To report an extremely rare case of ovarian borderline mucinous cystic tumor accompanied by low-grade endometrial stromal sarcoma (LGESS) with myxoid change.A 42-year-old woman complained of lower left abdominal fullness. Her serum carcinoembryonic antigen, cancer antigen (CA) 125, and CA19-9 levels were normal. Magnetic resonance imaging showed a 10-cm cystic mass with a 5-cm nodule in its wall, and a laparoscopy indicated a cystic mass at the left adnexa. Histology indicated a cystic lesion consisting of proliferative gastrointestinal-type epithelium; the mural nodule had a characteristic of striking myxoid change, preservation of arteriolar pattern, and a “tongue-like” infiltration.The diagnosis of ovarian mucinous borderline tumor accompanied by LGESS with myxoid change was appropriate. Mucinous cystic tumors of the ovary, whether benign, borderline, or malignant, may be associated with mural nodules of various types, including sarcomas, sarcoma-like mural nodules, foci of anaplastic carcinoma, carcinosarcoma, mixed nodules, and leiomyomas , 2. The A 42-year-old woman complained of lower left abdominal fullness. Her serum carcinoembryonic antigen (CEA), cancer antigen (CA) 125, and CA19-9 levels were within normal limits. Magnetic resonance imaging (MRI) showed a 10-cm cystic mass at the pelvic cavity with a 5-cm nodule in its wall. The laparoscopy showed a 10-cm cystic mass at the left adnexa. The right ovary and uterus were grossly normal. The patient underwent left salpingo-oophorectomy and adhesiolysis. The patient was symptom free 10 months after surgery.On macroscopic examination, the cyst measured 10 cm in maximal diameter and had a thickness of 0.2–0.5 cm. This cystic tumor focally adhered to the peritoneum. The inner side was slightly uneven and attached with clot-like material in some area. The cyst had a solid mural nodule protruding from the wall into the lumen measuring up to 5 × 4 × 4 cm in size (Fig. Microscopic observation showed that the cystic walls were lined mainly by single-layered tall mucinous columnar epithelium Fig. a. Some eA 5-cm mural nodule was identified in the hysterectomy specimen protruding into the cystic cavity, extending onto the serosal, and involving the tubal fimbria. The mural nodule had a characteristic of striking myxoid change, preservation of arteriolar pattern, and a “tonguelike” infiltration Fig. a. There Immunohistochemical staining is shown in Fig. Taking into account the above features, we made the diagnosis as ovarian mucinous borderline tumor accompanied by LGESS with myxoid change involving the tubal fimbria.In 1979, Prat and Scully  describeMural nodules can develop in benign, borderline, or malignant ovarian tumors. These mural nodules are seen as solid lesions on the wall of the tumor or protrude into the cyst lumen. The diagnosis of mural nodules in the residual ovary is based on the size and characteristic gross and microscopic features , 6. The A review of the literature revealed only five mucinous ovarian tumors with sarcomas, with most of the mural nodules measured > 5 cm in the greatest dimension. In these five cases, they were fibrosarcomatous mural nodule in an ovarian mucinous cystadenoma , fibrosaThe differential diagnosis for extrauterine LGESS depends on the location of the tumor. In case of ovarian location, ovarian sex cord–stromal tumors should be excluded. The pathological and morphological characteristics and immunohistochemical findings in the present case do not favor ovarian sex cord–stromal tumors. LGESS with myxoid change can be confused with myxoid smooth muscle lesions, but the “tonguelike” infiltration and small arterioles, along with the CD10 immunostaining and muscle marker negativity, will usually resolve the diagnosis.Tumors with a combination of different histology are divided into two clinicopathologic groups, collision or composite tumors . The colIn this case, there was a region of proliferation of large mononucleated cells just beneath the epithelium without forming a true nodule, which may help explain the occurrence of malignant mural nodules. We hypothesize the forming process of this sarcoma as follows. The submesothelial mesenchymal cells reacted to the intramural hemorrhage or the mucinous content of the cyst, which eventually became a hyperplastic nodule, probably corresponding to the earlier stage of the SLMNs. Then the proliferation of mesenchymal cells resulted in the sustained expansion of the lesion. The nodule enlarged gradually and became SLMNs. Some of the SLMNs could possibly evolve into a true sarcoma with the proliferation and differentiation of mesenchymal cells.We have presented a rare case of sarcoma-like mural nodule. Based on the ample evidence of the previous reports, and the immunohistochemical analysis of our case, we believe that this is a collision tumor with two different elements and may be formed by SLMN malignant change. Nevertheless, further molecular and genetic evidence is needed to support such a conclusion.


Primary bone lymphomas (PBLs) ,2 and ocA 55-year-old man presented with left hip pain without a history of trauma. His medical history and physical examination did not reveal any remarkable findings. The X-ray radiographs of the pelvis and left hip showed multiple lytic lesions. Body 18fluorodeoxyglucose positron emission tomography/computed tomography (18F-FDG PET/CT) demonstrated multiple osteolytic bone lesions in the left zygomatic bone, vertebral column, bilateral iliac bones, left caput femoris, and trochanter major of the femur with increased 18F-FDG uptake (SUVmax: 31) . Tru-CutConsidering the extensive lytic bone lesions and recent emergence of the OA tumor, the primary site of the disease must have been the bones in the presented case. Thus, the diagnosis can be categorized as PBL with OA involvement. An orbital mass was detected two months after diagnosis by means of MRI, but not by 18F-FDG PET/CT performed at diagnosis. It would be speculative to claim that such a large mass had arisen in a two month period. Taking into account that MRI is the gold standard imaging technique in evaluation of OA tumors , the orbEthics Committee Approval: Not applicable; Informed Consent: Not applicable.


Lipoblastomas are rare benign tumors that arise from embryonic white fat and almost always occur in babies and children. Here, we report a case of a giant popliteal lipoblastoma in a 23-month-old Japanese girl that was successfully treated via complete resection.Our patient was a 23-month-old Japanese girl. At 6 months of age, she presented at a nearby hospital with a mass on the popliteal side of her lower right leg. She had no symptoms and was diagnosed as having a benign adipose tumor via magnetic resonance imaging. The mass gradually increased in size, and she was referred to our hospital at 1 year and 11 months of age. A physical examination and radiology revealed a localized mass 13 × 10 × 7 cm in size in the aforementioned area that restricted knee movement and caused proximal tibia deformity. Magnetic resonance imaging showed a giant circumscribed subcutaneous mass with multiple partitions that was hyperintense on T1-weighted and T2-weighted images but not fat-saturated on T2-weighted images. Based on these findings, she was diagnosed as having a lipoblastoma. Because the mass surrounded her popliteal artery and vein and part of the popliteal nerve, surgical resection was considered risky, and we opted to simply observe her. However, owing to rapid growth of the mass and the worsening of symptoms, she underwent complete resection at 2 years and 6 months of age. A histological examination confirmed the diagnosis of a lipoblastoma. She was discharged from our hospital 3 days after surgery with no symptoms. She could walk without pain at the 6-month follow-up, and no local recurrence was observed.We successfully treated a giant popliteal lipoblastoma without complications by performing a total resection. Our report provides evidence that lipoblastomas should be considered for surgical resection when they progress or symptoms appear. Lipoblastomas (LBSs) are rare benign tumors, accounting for less than 1% of childhood neoplasms . ApproxiOur patient was a 23-month-old Japanese girl. She was born full-term after a normal pregnancy and delivery. At 6 months of age, she presented at a nearby hospital with a mass on the popliteal side of her lower right leg, which was diagnosed as a benign adipose tumor; the diagnosis was made via magnetic resonance imaging (MRI) because she had no symptoms. The mass gradually increased in size, and she had difficulty in walking.At 1 year and 11 months of age, she was referred to our hospital. A physical examination revealed a subcutaneous mass on the popliteal side of her lower right leg Fig. . MRI shoSurgical resection was considered but deemed risky and technically challenging because the mass surrounded her popliteal artery and vein and part of the popliteal nerve, as revealed by MRI. We decided to observe only, as the mass was not expected to grow. However, it grew rapidly Fig.  and her A histological examination of the tumor showed that it mainly consisted of mature fatty cells, fibrous partitions that created lobular cysts, immature lipoblasts, primitive mesenchymal cells, and stroma Fig. . These fAs defined by Jaffe in 1926, LBSs are atypical benign lipomatous lesions that consist of cells resembling embryonic white fat , 5. NeitOn radiography, an LBS appears only as a shadow of soft tissue, without calcification or specific features. MRI shows a lobulated fatty mass with various amounts of non-enhancing cystic areas and internal fibrous partitions intermixed with a few small enhancing soft tissue nodules . The hisLipomas do not contain internal fibrous partitions, which allows us to distinguish them from LBSs on MRI. Myxoid liposarcomas have internal fibrous partitions and myxoid areas as do LBSs; however, they occur in middle-aged and elderly patients, whereas LBSs mainly occur in children less than 3-years old.PLAG1 oncogene on chromosome 8q12 is rearranged in 70% of LBSs, and chromosome 8 polysomy is observed in 18% of LBSs [PLAG1 gene is involved in the development of the central neurological system [PLAG1 rearrangement for diagnosing LBS is 77%, and the specificity is 98% [Recently identified cytogenetic abnormalities may facilitate the diagnosis of LBSs. It is now clear that LBSs harbor a chromosome 8 rearrangement; the region containing the  of LBSs , 8–10. Tl system . The seny is 98% .The only treatment for LBS is complete surgical resection –5. No beWhen a patient with an LBS has no symptoms, the surgeon may decide to observe the patient rather than perform surgery. There are two reasons for doing so. First, surgery performed in young children is risky and requires considerable expertise. Second, LBSs are benign, and if their progression does not occur, they will likely become lipomas, with all immature lipoblasts becoming mature fatty cells. When the mass progresses rapidly during a prescribed period and functional symptoms appear, surgical treatment should be considered.After surgery, careful routine observation by MRI is required. Because tumors can recur as long as 84 months postoperatively, a follow-up period of at least 3 to 5 years is recommended, with MRI performed at fixed intervals [We successfully treated a giant popliteal LBS in a child without complications by performing a total resection. There was no local recurrence at the 6-month follow-up as assessed by MRI. LBSs are benign tumors that can progress rapidly and cause numerous symptoms without a spontaneous resolution. Therefore, if possible, surgical resection should be the treatment of choice for LBSs when the mass progresses or symptoms appear.


Anal melanoma is an aggressive but rare malignancy. Patients commonly present with very advanced or even metastatic disease. Risk factors for anal melanoma are family history and an activating mutation of C-KIT. Surgical excision remains the mainstay of therapy. The presence of activating mutations of C-KIT has prompted use of C-KIT inhibitors such as imatinib and sunitini. Early diagnosis and treatment remain crucial. Abdominal perineal resection (APR) offers a higher rate of local control whereas wide local excision (WLE) can yield superior long-term survival. Anorectal melanoma is an extremely rare malignancy with a high metastatic potential and equally high mortality rate . Of all Most patients afflicted with this malignancy present late in the course of the disease with non-specific complaints. In a case series of 18 patients, the most common presenting symptom was bright red blood per rectum . Due to A 79-year-old female with past medical history of diverticulosis, coronary artery disease, hypertension, varicose veins and diabetes, presented with bleeding per rectum for the last 4 months. The patient reported noting the blood while straining during a bowel movement. Few days prior to admission, the patient went to her primary care physician who diagnosed her with hemorrhoids and prescribed rectal suppositories. The patient reported that her problem persisted despite the suppositories. On the day of admission, she had sudden frank bleeding per rectum with severe abdominal and chest pain. She also felt a mass coming out along with the blood and tried unsuccessfully, to push the mass back in. She felt extreme pain which was associated with shortness of breath, lightheadedness and dizziness. She denied any fever, chills, blurry vision, cough, nausea, vomiting or diarrhea. She does not have any allergies or any family history of cancer. She denied any smoking, alcohol use, or drug use history.On physical exam, the patient was not in any acute distress with a PR 96/min, BP 110/52 mm Hg, temperature 98.6 °F, and RR 20/min. There was no pallor or jaundice present. Abdomen was soft, non-tender, non-distended, and bowel sounds present with no hepatosplenomegaly. Rectal exam revealed a dark colored mass measuring about 4 × 5 × 3 cm in size with a visible overlying blood clot. The mass was tender to palpation but had no active bleeding. Laboratory analysis showed Hb 11.8 g/dL, Hct 36.2%, WBC 8,400/μL, platelets 323,000,000/μL and comprehensive metabolic panel was completely normal. Chest X-ray was within normal limits.Patient was admitted for prolapsed hemorrhoids. Surgical consult was called and a CT scan of abdomen/pelvis was performed which showed colonic diverticulosis with no acute diverticulitis. The rectal mucosal appeared thickened compared with the remainder of the colon and it was advised that the possibility of a rectal or other colonic neoplasm be excluded . Biopsy Fig. 2-Anorectal melanoma is the third most common melanoma and it is the most common primary melanoma of the gastrointestinal tract . Risk faPrompt diagnosis of anorectal melanomas is critical due to its highly aggressive nature. A mass may be identified on sigmoidoscopy or colonoscopy but is often initially misdiagnosed as other anorectal malignancies . ComputeDue to its rare nature, there is a lack of evidence from randomized control trials regarding appropriate treatment of anorectal melanomas . HistoriThe role of medical therapy in the treatment of anorectal melanomas is another topic of debate. Little data exist to suggest its effectiveness; however, as more is uncovered about the pathophysiology of the malignancy, targeted therapies are being utilized. By far the most commonly used chemotherapy agent is dacarbazine . ImmunotDespite excision and adjuvant radiotherapy, anorectal melanomas carry a bleak prognosis. This is contributed in part to the non-specific complaints that patients present with and the high propensity of metastasis. In a patient case series of anorectal melanoma, the most common sites of metastasis were to the liver, lungs and pelvis . PrognosA heightened awareness of the presentation of anorectal melanomas amongst practitioners will be critical to more prompt diagnoses and improved clinical outcomes. Further research is also indicated to better delineate appropriate therapeutic course of this silent malignancy.


Malignant mesenchymoma (MM) is defined as a heterogeneous malignant soft tissue tumor that consists of 2 or more distinctly different mesenchymal components in addition to fibrosarcomatous elements. Bladder MM was rarely reported in the literature and there are only 5 cases of primary bladder MM documented in English literature to date.A 58-year-old male complained of difficulty in urination and intermittent gross hematuria for 3 months. Doppler ultrasound scan revealed an avascular and homogeneous hypoechoic mass measured 6.5 × 9 cm in the bladder. Computed tomography showed a homogeneous solid mass in the bladder.Pathology revealed spindle-shaped tumor and proliferation of poorly differentiated immature mesenchymal cells rich in eosinophilic cytoplasm with hyperchromatic sticklike nuclei. Immunohistochemical examinations were positive for CD117.The patient was diagnosed with presence of bladder tumor and underwent radical cystectomy; the optimal treatment strategy was reviewed and discussed.There was no recurrence or metastasis during a 16-month follow-up.Our case study demonstrated bladder MM with a relatively indolent clinical course. A multidisciplinary approach including surgery, radiotherapy, and chemotherapy may be useful. Radical resection is the most important determinant of clinical outcome. Generally, the clinical outcome and prognosis of mesenchymoma are favorable. It was defined as a malignant soft tissue tumor that consists of 2 or more distinctly different mesenchymal components. Primary MM of bladder was reported occasionally; a review of the literature revealed only 5 cases fit the criteria of primary bladder MM.–6 To our knowledge, the present case is the sixth case in the English literature to date.Malignant mesenchymoma (MM) is a rare tumor and was first described by Stout in 19482The patient was a 58-year-old male who complained of difficulty in urination and intermittent gross hematuria for 3 months. He was admitted to our hospital on June 1, 2010. There was no family history of cancer or chronic illness. Rectal examination disclosed a mildly enlarged mass just like benign prostate. Abdominal exploration was unremarkable. Urinalysis revealed gross and microscopic hematuria though urine Gram stain did not suggest infection. Biochemical investigations revealed mild hyperbilirubinemia and the rest parameters were normal. Urinary cytology studies showed no abnormalities. Laboratory findings showed blood counts, electrolytes, and other routine studies were all normal. The preoperative Doppler ultrasound scan revealed an avascular and homogeneous hypoechoic mass measuring 6.5 × 9 cm in the bladder. A subsequent computed tomography (CT) demonstrated a large mass showing homogeneous density in the bladder region without obvious enlarged lymph nodes , as well as mesothelioma and other rare sarcomas. There is corresponding relationship between the size of the tumor and cells atypia and the prognosis of MM. Immunohistochemical studies were necessary to establish the diagnosis.MM arises from a primitive mesenchymal cell with the capacity for totipotent differentiation. It can occur at all locations in the body, especially in the retroperitoneum, soft tissue, and digestive tract such as gastrointestinal stromal tumors (GIST), which are predominant in adults or malignant extra-GIST, which are very rare. including the MM. Radiology and/or chemotherapy resulted in a differential response. Randomized clinical trial illustrated that targeted treatments such as high-dose Imatinib can improve progression-free survival in gastrointestinal stromal tumors. In our case, the clinical course was rather indolent. Although without any adjuvant chemotherapy or radiotherapy, there was no evidence of metastasis during the subsequent 16-month period. The patient was in good health; it seems to be a more favorable prognosis.Given its scarcity, there are insufficient data to suggest the optimal management strategy for MM of the urinary bladder. A multidisciplinary approach including surgery, radiotherapy, and chemotherapy may be useful. Radical resection is the most important determinant of clinical outcome. Chemotherapy and radiotherapy were ineffective for the soft parts of the sarcoma, The other just received radical cystectomy and died 2 months after diagnosis. The prognosis of MM is poor. With few exceptions, the median survival time is about 30 months from diagnosis.In the 3 available documented bladder MMs, sizes were 18 × 10 × 9, 14 × 13 × 7, and 14 × 13 × 7 cm, respectively. It seems to be larger than traditional bladder transitional carcinoma (BTCC). Among these 3 cases, only 2 have detailed follow-up data. One patient received surgery, chemotherapy, and irradiation, and had a total life of 21 months.4 Patients less than 40 years old or those who had an RMS component showed significantly worse prognosis; the other mesenchymal components showed no significant correlation with survival.The prognosis of MM remains controversial. There is no statistically significant prognosis indicator with respect to gender, tumor site, tumor size, or MIB-1-labeling index.


Synovial chondromatosis is characterized by cartilaginous metaplasia in synovial tissues. Extra-articular tenosynovial chondromatosis is considered to be an anatomical counterpart of articular synovial chondromatosis. Extra-articular tenosynovial chondromatosis occurs preferentially in the hand, although its frequency is low.We report three cases of extra-articular tenosynovial chondromatosis. A 65-year-old female presented with a history of symptoms over 40 years related to the dorsum of her index finger (Case 1), A 46-year-old female presented with a 6-month history of symptoms at the volar surface of her middle finger (Case 2), and a 66-year-old male presented with a 3-month history of symptoms in a dorsal ring finger. Case 2 had evidence of ossification, which could be classified as osteochondromatosis. Interestingly, the index finger lesions (Case 1) were accompanied by excessive bone involvement. The signal intensity of T2-weighted magnetic resonance imaging varies from low to high, possibly reflecting histological variations, such as ossification and fatty tissue changes. All lesions were resected without complications.Variations in anatomical sites suggest that overuse or mechanical overloading was not causative. Extensive involvement of the nearby tendon and joint capsule, as well as the bone, would require attention during the resection. Preoperative analysis of images is important, not only for the diagnosis, but also to assess the extent of the lesion, particularly given the complex anatomy of the finger. Synovial chondromatosis is an unusual condition that is characterized by cartilaginous metaplasia in the synovial membrane of joints, bursae, and tendon sheaths -3. ArticExtra-articular tenosynovial chondromatosis of the finger is a rare condition. Signs and symptoms include swelling, reduced range of motion, and pain . Herein,1. Two females and one male patient  were among the three cases of extra-articular tenosynovial chondromatosis of the finger. All cases were right-hand dominant. The male patient was retired, and the other 2 female patients were housewives. There was no history of trauma, repetitive stress, and/or over usage of the fingers among the cases. The lesions were located on the dorsal side in two cases, and the volar side in one case. Two of the lesions were located under the tendon, and for the remaining case the lesion was above the tendon. Multiple nodules were observed in two cases, whereas a singular nodule was present in one case. Each nodule was 1 cm to 2 cm in size. Intraosseous invasion was seen in the case with calcification. Adjacent degenerative changes were not observed in any cases. The duration from onset varied; it was 40 years, 6 months, and 3 months in these cases. The lesions were treated by curettage. We describe the details of each case below.Clinical data are summarized in Table 1a). A magnetic resonance image (MRI) showed multinodular lesions under the extensor tendon with homogeneous intermediate signal intensity on the T1-weighted images and heterogeneous low to high signal intensify on the T2-weighted images. The lesions extended from the PIP joint to the MP joint on the dorsal side. Osseous invasion was evident . The resection was performed under general anesthesia, with a tourniquet at the upper arm, through a zigzag incision on the dorsum of the index finger, from the PIP joint to the MP joint. The presence of a white colored lesions covered by synovial tissue was confirmed before each resection. At the PIP joint the capsule was connected to a lesion, but the lesions did not extend into the joint. The involved extensor tendon was preserved, although parts of the lateral hood and joint capsule were resected with the lesion. Lesions in the bone at the distal proximal phalanx and the distal metacarpal were curetted. Histologically, the lesion is composed of multiple nodules of hyaline cartilage containing chondrocytes arranged in clusters and separated by fibrous septa. Synovial lining was visible covering the cartilaginous nodules. The features are compatible with synovial chondromatosis . Malignant or infectious features were absent. Two years after the operation, a follow-up physical examination confirmed no recurrence of a lesion. A slight reduction in the range of motion in the PIP joint was still evident, but there was no effect on the activities of the daily life.A 65-year-old female presented with complaints of a painless but hard irregular swelling on the dorsum of her index finger. She described the swelling as having slightly increased in size over the 40-year period since onset, but had noticed more of an increase over the previous 2 months. There was no history of trauma or overuse. Despite experiencing that a full grip was difficult; we noted a slightly reduced range of motion in the proximal interphalangeal (PIP) joint, no restrictions in her daily activities were reported. Multiple lesions comprising hard masses, some of which had elastic properties, were palpable over the dorsum of the PIP joint and metacarpophalangeal (MP) joint of the index finger. Plain radiographs of the index finger showed evidence of calcification. Irregularity in the adjacent bone surfaces was observed . An MRI revealed a lesion located above the flexor tendon. The signal intensity of the lesion was heterogeneous with low to high signal intensity on the T1- and T2-weighted images. Foci of high signal intensity were also observed in the T1- and T2-weighted images, suggesting the presence of fatty tissue . A resection was performed under general anesthesia, with a tourniquet applied at the upper arm. A curved incision was made starting at the base of the radial side of the index finger and extending over the lesion at the proximal phalanx. After confirming the presence of a white colored hard lesion, it was dissected from the surrounding tissue. Histologically, the lesion was composed of osteocartilagenous tissue, with irregularly thickened bone trabeculae and fatty marrow. The lesion was surrounded by fibrovascular connective tissue. The features are compatible with synovial chondromatosis . Malignant or infectious features were absent. No recurrence was observed at the time of follow-up 11 months after the operation. Full range of motion was restored without any effect on activities of daily living.A 46-year-old female complained of swelling in her right middle index finger that she had endured for 6 months. The range of motion of this finger was slightly reduced in flexion because of the mass of a lesion. There was no history of pain or trauma. The patient reported no tenderness over the area of the lesion. Plain radiographs of the hand showed a small osseous node in the middle finger at the radial flexor side . An MRI showed multinodular lesions located at the dorsal aspect of the proximal phalanx under the tendon. The lesions had homogeneous intermediate signal intensity on the T1-weighted images and heterogeneous low to high signal intensity on the T2-weighted images . The resection was performed under general anesthesia, with a tourniquet applied at the upper arm, and a mid-dorsal incision was made. Parts of the lateral hood were resected along with the lesion. Histology showed that the lesion was composed of multiple nodular or lobular structures of what appeared to be mature hyaline cartilaginous tissue, surrounded by synovial tissue (not shown in figure), the features of which were compatible with synovial chondromatosis. No signs of malignancy or infection were observed. A follow-up performed 5 years and 2 months after surgery confirmed no recurrence of a lesion. Full range of motion was maintained without any complications.A 66 year-old male complained of a painless swelling on his right ring finger that he had noticed for 3 months. There was no limitation in the range of motion of the finger, although the lesion caused discomfort in full grip. There was no history of trauma or overuse. On physical examination, the mass of the lesion was palpable at the dorsum of the right finger at the distal of the MP joint. The plain radiographs revealed no apparent abnormalities. No calcification was observed  . Of thesFor articular synovial chondromatosis in the hip and knee, although the risk of malignant transformation is small, resection of the cartilaginous lesions and synovectomy tend to be recommended . In contIn the current series, the recurrence of lesions was not observed during the follow-up periods. The overall recurrence rate for articular chondromatosis ranges from 3% to 23% , 7. In cThis case series study reports three cases of extra-articular tenosynovial chondromatosis of the fingers, including one case with excessive bone involvement. There were no specific determinants for the onset of the pathology identified among these cases. The variation in anatomical site suggests the cause was not associated with overuse or mechanical overload. The MRI findings reflect the histological variation among cases. Extensive lesions with tendon or bone involvement add to the anatomical complexity, making preoperative image analysis imperative, not only for the diagnosis, but also when planning the resection.


Mobile schwannomas of the spine have been sparsely documented in the literature. In cases referred to in existing literature, the migratory schwannoma was documented to occur in the lumbar spine. We added another case to the small available literature. In our case report, the patient had a previously known lumbar schwannoma that was being managed conservatively. Due to an acute change in clinical symptoms, repeat imaging was performed. A magnetic resonance imaging (MRI) of his spine revealed migration of the schwannoma two levels rostral to his recent imaging from six weeks earlier. The patient underwent surgical resection of his lesion. During the operation, the ultrasound was utilized to confirm the lesion prior to dural opening. In this report, we attempt to provide further evidence of the utility of an intraoperative ultrasound for intradural lesions and intend to add to the published literature of mobile schwannomas of the spine Although rare, mobile schwannomas have been observed at various vertebral levels. Most of them have been reported within the lumbar spine. As described in the available literature, preoperative imaging confirmed the level of the operative intradural lesion. In several instances, imaging was performed just several hours prior to the operation. In a surgical nightmare, operative findings revealed normal spinal anatomy at the previously determined position -4. RegarA 40-year-old gentleman presented with a history of progressive lower back pain and circumferential right leg pain. Over a period of one week, the pain developed into higher low back pain along with bilateral lower extremity weakness and increasing difficulty with balance. Previously, a magnetic resonance imaging (MRI) of his thoracolumbar spine demonstrated an intradural, extramedullary mass in the T11 to T12 intradural region.A repeat MRI was performed due to the new onset bladder incontinence. Upon further review of his most recent imaging, the lesion was located at T12-L1, no longer as high as T11 /MRI confirmed cases reported to date, this one included. Table When comparing preoperative tumor location and final tumor position upon excision, each case demonstrates a significant change in the vertebral level of the lesion. Primary factors driving tumor movement include postural adjustments and arrangement on the operating table as well as positive pressure respiration and other actions that influence intrathecal, intrathoracic, or intra-abdominal pressures such as the Valsalva maneuver , 7, 9-10The enhanced risk of surgically removing a mobile schwannoma comes not from dural attachment as almost all cases reported no dural adhesion. Instead, they arose from an unpredictable and often substantial movement of these lesions up until the moment of excision , 9. WithAn intraoperative ultrasound has proven very effective in tumor localization before entering the dural space and does not significantly increase surgical risk or procedure length . AdditioMigratory schwannomas of the spine are rarely reported in the literature. We add to the available literature another case of a mobile lumbar schwannoma. Due to its location at the cauda equina, a schwannoma in this region may be more prone to migration. An intraoperative ultrasound is essential in confirming operative location prior to dural opening. Also, we present a brief review of the available literature regarding this unusual phenomenon.


Since the concept of intraductal tubulopapillary neoplasm (ITPN) was introduced in the current World Health Organization classification of tumors, the number of reports of ITPN occurrence has increased gradually. However, ITPN is usually located in the main pancreatic duct, with few reports of a branch duct ITPN. As a result, imaging protocols for the diagnosis of a branch duct ITPN have not been established.We report a case of a concurrent presentation of a branch duct ITPN and intraductal papillary mucinous neoplasm (IPMN) in the head of the pancreas, with a superior mesenteric artery (SMA) aneurysm. Initially, the cystic masses in the pancreatic head were diagnosed as branch duct IPMNs, with treatment consisting of a pylorus-preserving pancreaticoduodenectomy, in combination with an aneurysmectomy performed for treatment of the SMA aneurysm. Pathological examination confirmed these cysts were a combination of branch-type ITPN and IPMN. The patient recovered from the treatment without complication, with no evidence of recurrence over a period of 34 months post-surgery.This case report of a synchronous presentation of an ITPN and IPMN indicates the difficulty in differentiating these two types of neoplasms in the branch duct of the pancreatic head. Cystic lesions in the pancreas have been identified with increasing frequency in recent years due to the development and advancement of imaging technology. Although some of these cystic tumors, such as intraductal papillary mucinous neoplasms (IPMNs), have a potential for malignancy that cannot be ignored, considering the risks associated with pancreatic resection, surgical treatment must be considered with caution. Therefore, accurate imaging is clinically important to establish a diagnosis and indication for surgery.Intraductal tubulopapillary neoplasms (ITPNs) were introduced as a new type of pancreatic tumor by Yamaguchi et al. in 2009  and subsAn aneurysm of the superior mesenteric artery (SMA) is also a rare occurrence, accounting for only 6.9% of all visceral artery aneurysms , and carA 55-year-old woman presented to a local hospital complaining of epigastralgia and anemia. Endoscopic examination of the upper gastrointestinal tract revealed gastritis, with pancreatic masses observed on abdominal ultrasonography. Computed tomography (CT) imaging further revealed an aneurysm of the SMA. Based on these findings, the patient was admitted to our hospital for further investigation and treatment.Her medical history included hypertension, cerebral hemorrhage, and panic disorder, with no indication of hyperglycemia. Findings on physical examination were unremarkable and laboratory data, including tumor markers, were within normal limits. Dynamic CT imaging revealed a saccular SMA aneurysm, with a 2.4-cm diameter, located 2 cm distal to the origin of the SMA Fig. –c, as weAfter obtaining informed consent, we simultaneously performed a pylorus-preserving pancreatoduodenectomy and SMA aneurysmectomy. Macroscopic examination revealed the presence of two cystic masses in the pancreatic head. Neoplastic cells were identified in one of these masses, characterized by enlarged nuclei and eosinophilic cytoplasm containing little mucus Fig. –c. The oThe patient’s postoperative course was uneventful and her length of hospital stay was 35 days. She has remained symptom-free since treatment, with no recurrence of the neoplasm over the 34-month follow-up.It has been reported that ITPN accounts for only 0.9% of all pancreatic exocrine tumors and 3% of all pancreatic intraductal neoplasms . AlthougInitially, we had diagnosed the two cystic masses as IPMNs. According to the international consensus guidelines for the management of IPMNs (2012), immediate resection is not indicated  for branch duct IPMNs with a diameter > 3 cm and without observable mural nodules . In the Histologic examination of the two cystic tumors identified on preoperative imaging confirmed the presence of two types of neoplasms, namely an ITPN and an IPMN. Yamaguchi et al. described the pathological characteristics of an ITPN as follows: 1) a solid nodular tumor obstructing dilated ducts on macroscopic examination, (2) no visible secretion of mucin, (3) tubulopapillary growth, (4) uniform high-grade atypia throughout the neoplasm, (5) an easily recognizable necrotic foci, (6) ductal differentiation, and (7) absence of acinar differentiation  a solid .In general, the prognosis for ITPNs is considered to be much better than the poor prognosis for pancreatic ductal adenocarcinoma , 16. TheWith respect to SMA aneurysms, surgical interventions including pancreatic resection and stent-grafting are recommended treatments due to the risk of rupture . In our We identified 44 articles published in English addressing ITPN in PubMed using the keywords “ITPN” and “intraductal tubulopapillary neoplasm,” as well as four Japanese articles from other sources. Eight articles addressing ITPN in the bile duct were excluded, while another 10 articles were excluded because they did not provide detailed patient data. Therefore, we extracted comprehensive data on 51 cases of ITPN from 26 articles , 17–36. Therefore, our case of a concurrent branch duct ITPN and IPMN in the pancreatic head, with a SMA aneurysm, is a very rare occurrence, with an association between an ITPN and IPMN not previously having been reported. Our case underlines the difficulty in differentiating these two types of neoplasm pre-operatively. Therefore, an ITPN should be included in the differential diagnosis, even when a cystic mass suggestive on an IPMN is observed.


MRI is highly valuable to characterize cysts.Pancreatic polycystosis is one of rare causes of recurrent abdominal pain of pancreatic origin in children frequently associated with other organ's cysts which are to be searched. Association with pineal cyst is exceptional, and link between the two locations is to be elucidated.  Polycystic pancreatic disease is a rare condition in adults and is usually found incidentally during surgery or at autopsy A 14‐year‐old female Caucasian adolescent, in whom two cystic images (15 and 10 mm) in the pancreas were discovered on abdominal US, was referred to the pediatric GI OPD of Dreux hospital (France). She suffered from isolated upper abdominal pain for 10 days. She had an unremarkable physical examination. Her weight was 45.4 kg, height 148 cm, and her pubertal stage was Tanner 5. The parents were not consanguineous. There was no family history of disease of pancreas, kidney, liver, or cystic disease. As relevant past history, she had presented with upper abdominal pain which had lasted few days a year earlier. Upon admission in the OPD, she was free of symptoms. A treatment with a proton pump inhibitor was prescribed by the family physician., and C peptide values were normal as well as oral glucose tolerance test. Abdominal MRI revealed large number of pancreatic cysts of variable sizes throughout the parenchyma, with the biggest measuring 20 mm in diameter , retentional, tumoral, parasitic, pseudo, or duplication cysts Abdominal MRI was of tremendous contribution to the diagnosis by revealing the number of cysts, with a yield that, was superior as it provided an excellent tissue characterization along with detailed anatomical definition of the pancreas. Pancreatic cysts require no treatment, except for symptomatic one. It is rare that surgery is required. In that case, several modalities are reported, such as coelioscopic fenestration of big ones Pineal cysts are frequently seen on brain MRIs in children with a prevalence which changes with age, more frequently in girls and with a peak in young adults found to be 2–4% of brain MRIs Polycystic disease of the pancreas being very rare in children, the literature lacks information on its prevalence, etiopathogenesis, management, appropriate follow‐up modalities, and long‐term outcome. To our knowledge, association with pineal cyst was not previously reported.A‐MB (GI pediatrician consultant for whom the patient was referred): conducted the work‐up until the definitive diagnosis, redacted the discussion and the key clinical message, and participated in the bibliography research. AF: redacted the case history and participated in the preliminary form of the manuscript. SKK (Pediatric consultant): participated in the bibliographic research selection, in the discussion, in conclusion, and in the manuscript review.No conflict of interests to be declared by the authors.


A 58-year-old-man presented to the emergency unit with abdominal pain and fever for three days. He had no history of nausea or vomiting. The abdominal examination revealed a minimally distended abdomen with tenderness in the right lower quadrant (RLQ). At palpation, there was no rebound tenderness or defence. Laboratory findings revealed inflammation and a high white blood cell count, predominantly neutrophils.Contrast enhanced CT examination of the abdomen shows a 12cm, oval-shaped, low-attenuation cystic lesion (21 HU) with mural curvilinear calcifications  was made. Surgery revealed the presence of greyish white gelatinous material into the peritoneal cavity confirming the diagnosis of rupture of a mucocele. Open laparotomy was chosen in order to minimize trauma and avoid further spilling of mucus. The gelatinous material was removed and the mucocele of the appendix was resected.AM is a progressive dilatation of the appendix from intraluminal accumulation of mucoid substance . It may PMP is difficult to treat and carries an uncertain prognosis, whether the mucocele originates from benign or malignant disease. CT is considered as the most accurate method of diagnostics and can be used to discover the signs specific to mucocele with a high accuracy .In our case, the histopathological examination confirmed the diagnosis of low-grade appendiceal mucinous neoplasm (mucinous cystadenoma). A specimen of the intra peritoneal mucus showed no evidence for presence of epithelial cells. The patient was referred to a specialised centre to perform further follow-up.Although a mucocele is encountered in less than 0.5% of patients undergoing appendectomy, accurate preoperative diagnosis is important as it may reduce the possibility of rupture and peritoneal contamination during resection. It is the responsibility of the radiologist to alert the surgeon on the presence of such an entity before surgery.The authors declare that they have no competing interests.


Mitotically active cellular fibroma (MACF) of the ovary, characterized by relatively high mitotic activity without severe atypia, represents a relatively new disease entity. MACF is categorized as a benign ovarian tumor. However, due to a limited number of case reports, its clinical and pathological features and optimum management remains largely undetermined. Herein, we report on a rare case of MACF that grew rapidly in size and was diagnosed on detailed pathological examination.FOXL2 mutation analysis excluded the possibility of an adult granulosa cell tumor, and the patient was diagnosed with a MACF of the ovary.A 44-year-old Japanese woman, who detected a myoma-like lesion 1-year earlier, was referred to our hospital when the follow-up examination demonstrated that the mass had increased in size. Magnetic resonance imaging revealed a T1 isointense and T2 hyperintense tumor (11 cm in diameter) in the right pelvic cavity. Laparoscopy confirmed the presence of a right ovarian tumor and laparoscopic right adnexectomy was performed. The tumor cells consisted of dense cellular proliferations of spindle fibroblast-like cells without significant cytological atypia. The mitotic activity index was estimated at >15 mitotic figures per 10 high-power fields. Reticulin staining and To the best of our knowledge, we are the first to report on a case of rapid growth of a MACF of the ovary during follow-up. When an increase in the size of a solid ovarian mass is detected, a MACF should be considered as a differential diagnosis. Ovarian fibrous tumors were previously classified as benign fibromas or malignant fibrosarcomas, according to the World Health Organization’s classification of tumors of the breast & Female Genital Origans . OvarianHerein, we report on the clinical and histopathological characteristics of a rare case of rapid growth of a MACF of the ovary that almost doubled in size during a 1-year follow-up period and provide a review of the literature.A 44-year-old Japanese woman underwent a medical examination for health check reasons at her local hospital. An ultrasound scan revealed a uterine myoma-like lesion, 5.9 cm in diameter. The patient revisited the hospital 12-months later for a follow-up examination. In that time, the lesion had increased to approximately twice its size. The patient was referred to our clinic for a detailed examination. At the first internal examination, the body of the uterus was enlarged to the size of a newborn head; cervical and vaginal discharge was unremarkable. The adnexa were not palpable on both sides. The patient’s blood test results were normal. Transvaginal ultrasonography detected an isoechoic solid mass with an ill-defined boundary between the lesion and the uterus Fig. . A tumorMacroscopically, the cut surface of the tumor was homogeneous and solid in appearance with a yellowish-white pigment. Necrosis and hemorrhage were absent within the tumor Fig. . MicroscFOXL2 c.402C > G (p.C134W) point mutation that is observed in the majority of AGCT patients [FOXL2 mutation was detected in the AGCT case used as a positive control, but was absent from our case and the negative control  and mild to moderate nuclear atypia . CellulaFOXL2 c.402C > G (p.C134W) point mutation is detected in 95.0 % of AGCT patients, especially those with the diffuse type [FOXL2 point mutation analysis and reticulin staining can be employed in differential diagnosis of cellular fibroma and AGCT. In this case, we rejected a diagnosis of AGCT based on FOXL2 mutation analysis and reticulin staining, and confirmed a final diagnosis of MACF.The use type –20. It hWe report our experience of a case of rapid growth of a MACF of the ovary during a 1-year follow-up period. When preoperative serum tumor marker levels and/or imaging findings do not suggest ovarian cancer in instances of rapid growth of a solid ovarian mass, MACF should be considered as a differential diagnosis. Our case and review of the literature suggest that MACF of the ovary may represent a heterogeneous disease entity that requires an accumulation of case reports to establish diagnosis and treatment.


To explore effective treatment of large abdominal malignancies in children complicated with abdominal compartment syndrome (ACS).Six children with large abdominal malignancies complicated with ACS were admitted to our department from January 2013 to January 2016, and the changes in their breathing, heart rate, oxygen saturation, abdominal circumference, bladder pressure, and urine output, as well as the treatment measures and outcomes, were retrospectively analyzed.The 6 children included 1 child with bilateral nephroblastoma, 1 child with abdominal alveolar rhabdomyosarcoma, 1 child with right ovarian malignant teratoma complicated with abdominal glioma, 1 child with abdominal malignant teratoma, 1 child with right nephroblastoma, and 1 child with left adrenal gland neuroblastoma. All patients were treated in a timely manner. The first 4 children underwent abdominal cavity decompression through surgical resection of the tumor, and the ACS was successfully cured allowing for follow-up care, whereas the last 2 patients failed to receive emergency surgery and eventually died due to the gradual aggravation of ACS.Decompression through surgical resection of the tumor is the only effective measure for treating large abdominal malignancies in children complicated with ACS. Clinical risk factors for the increase in IAP include extensive burns, massive rehydration, pancreatitis, mechanical intestinal obstruction, post-abdominal-closure under tension, postoperative bleeding, retroperitoneal hemorrhage, and abdominal tumor. In particular, children with large abdominal malignancies show no obvious early symptoms, and most are unable to complain about the physical discomfort. However, the tumors are generally large when diagnosed and developed rapidly, leading to a sharp increase in IAP that is prone to be complicated with ACS. Clinically, the condition is a rare emergency situation that has a high mortality rate. In the past 3 years, 6 children with large abdominal malignancies complicated with ACS were admitted to our department, and the details of their cases were analyzed and reported in this paper.Abdominal compartment syndrome (ACS) refers to the systematic dysfunction of multiple organs, including the heart and blood vessels, lung, kidney, stomach and intestine, and the brain, resulting from a marked increase in intra-abdominal pressure (IAP) from any cause.22.1From January 2013 to January 2016, 6 children suffering from large abdominal malignancies complicated with ACS were admitted to our department, including 3 males and 3 females aged 1 month to 3 years old with a mean age of 19 months old. The children weighed 5 to 13.5 kg, with a mean of 10.5 kg. The 6 children included 1 child with bilateral nephroblastoma, 1 child with abdominal alveolar rhabdomyosarcoma, 1 child with right ovarian malignant teratoma complicated with abdominal glioma, 1 child with abdominal malignant teratoma, 1 child with right nephroblastoma, and 1 child with left adrenal gland neuroblastoma. All children showed symptoms and signs including shortness of breath, obvious abdominal distension, and fast heart rate upon admission.2.2After admission, the children were immediately given life support such as oxygen and fluid resuscitation, and vital signs and blood oxygen saturation were monitored. A urinary catheter was placed, urine output was monitored, and bladder pressure was measured. The presence of respiratory, circulatory, and urinary system dysfunction was assessed, and corresponding symptomatic treatment was started. Once ACS was diagnosed, abdominal decompression was immediately performed using surgical methods based on the specific circumstances of the child. A longitudinal or transverse incision of the abdominal wall was created, then the tumor was removed, and staged closure of the abdominal wall was performed.2.32O = 0.74 mm Hg.,5In our department, intravesical pressure was measured to indirectly determine the IAP. The specific methods were as follows. The children were placed in the supine position with a Foley catheter inserted to empty the bladder. A transparent extension tube was added and connected to a 3-way connector. The catheter and the extension tubes were lifted to inject saline into the bladder with injection volumes of 1, 2, and 3 mL/kg with a maximum volume of 50 mL. The midaxillary line was used as the baseline; the vertical distance between the baseline and the lowest point of the liquid level was measured with a scale, and the values were recorded when no significant difference was present between the 2 measured values. These values were converted into IAP measurements according to the formula, 1 cm H2.4The bladder pressure, respiration, heart rate, oxygen saturation, and urine output of the 4 children who underwent surgery were collected 6 hours before and 12 hours after surgery for analysis, and the abdominal circumference was compared before and after surgery.3All of the 6 children suffered from large abdominal malignancies that had progressed after admission. These conditions were diagnosed as large abdominal malignancies complicated with ACS.3.1The first child suffered from bilateral nephroblastoma. After admission, a bilateral tumor biopsy was performed under ultrasound guidance, and neoadjuvant chemotherapy was planned after obtaining the pathological results. However, during the waiting process, the child showed gradually aggravated abdominal distension, poor mental reaction, shortness of breath, a rapid pulse, a decrease in blood oxygen saturation to 80%, and oliguria, with an abdominal circumference of 53 cm and IAP of 25 mm Hg. Because conservative treatment achieved poor results, resulting in a prolongation of the critical condition, surgery was immediately performed to remove the left nephroblastoma, followed by right renal tumor biopsy. After 2 courses of postoperative chemotherapy, the right renal tumor shrank, followed by a compensatory increase of the normal kidney tissue. Then, radical resection of the right nephroblastoma  was performed, with good recovery. After surgery, the abdominal circumference was 42 cm, and the IAP was 5 mm Hg. Follow-up chemotherapy was initiated Fig. .3.2In this child suffering from abdominal embryonal rhabdomyosarcoma, neoadjuvant chemotherapy was planned first. On the day we started chemotherapy, the condition of the child deteriorated, the breathing and heart rate increased significantly, and the blood oxygen saturation decreased to 75%, with an abdominal circumference of 65 cm and IAP of 30 mm Hg. Because the condition was life-threatening, surgery was immediately performed, and the intraoperative findings showed that the tumor occupied almost the entire abdominal cavity, wrapping around the left iliac blood vessels and left ureter. Therefore, atypical resection (leaving part of the tumor in place) of the large abdominal rhabdomyosarcoma was performed, combined with left ureter anastomosis and cystostomy. After surgery, the IAP decreased to 8 mm Hg, the abdominal circumference was reduced to 43 cm, and vital signs could be maintained. Regular chemotherapy was conducted for 11 weeks after surgery, and then a secondary surgery was performed. Retroperitoneal rhabdomyosarcoma resection and abdominal wall tumor resection, followed by regular chemotherapy, with no radiation therapy was used. One year and 10 months after surgery, tumor recurrence was found, and the operation was performed again. The intraoperative findings showed that the tumor surrounded the left external iliac artery and vein. After the left external iliac vein was truncated, the tumor was completely removed, and then anastomosis of the left external iliac vein was performed Fig. .3.3One child had a right ovarian malignant teratoma complicated by gliomatosis peritonei. Before admission, computed tomography at another hospital suggested massive abdominal effusion, right pleural effusion, and pericardial effusion. After admission, an abdominal tumor biopsy was performed under ultrasonographic guidance. Alpha-1-fetoprotein was 380.7 ng/mL. Then, the child showed progressive abdominal distension, impairment of respiration, poor appetite, oliguria, and the blood oxygen saturation decreased to 88%, with an abdominal circumference of 60 cm and IAP of 26 mm Hg. The condition was diagnosed as ACS. Surgery was performed to remove the right ovarian large teratoma, omentum, and right subphrenic tumor. After surgery, the blood oxygen saturation and IAP returned to normal levels, with an abdominal circumference of 41 cm. Alpha-1-fetoprotein was 1.5 ng/mL. The clinical condition of the child improved, and we were able to start chemotherapy : if the tumor is not complicated by ACS, neoadjuvant chemotherapy is started first, and then surgical resection is performed after the tumor volume has decreased. If the tumor is complicated by abdominal compartment syndrome, surgical abdominal decompression is conducted immediately. This strategy prevents further compression of the inferior vena cava and the portal vein. During chemotherapy, tumor necrosis or intratumoral hemorrhage resulting in increased volume of the tumor in the short term can also aggravate the process of ACS. For the second case in this paper, a child who suffered from abdominal alveolar rhabdomyosarcoma complicated with ACS, neoadjuvant chemotherapy was performed first, but on the day that chemotherapy was started, abdominal distension worsened, and the blood oxygen saturation deteriorated. Therefore, laparotomy decompression was immediately performed, and the presence of tumor necrosis and intratumoral hemorrhage was confirmed intraoperatively. The families of our fifth and sixth patients refused to accept surgical treatment, and ACS continued to intensify, causing irreversible dysfunction of multiple organs and ultimately leading to death. Therefore, for large abdominal malignancies in children complicated with ACS, after obtaining the consent of the patient's family, laparotomy decompression should be performed immediately.Large abdominal malignancies in children are frequently characterized by the following parameters: rapid progression, generally large tumor upon diagnosis, rich blood supply to the tumor, and tumor tissue surrounding vital organs or blood vessels. As a result, heavy bleeding that is difficult to control often occurs intraoperatively, and the tumor cannot be completely removed through surgery. During the resection of large tumors, life-threatening incidents may occur. In addition, the previous consensus for treating large abdominal malignancies in children was to conduct neoadjuvant chemotherapy first and then to perform surgical resection of the tumor after the tumor volume had decreased. Because of the above limitations, controversy still exists regarding the priority of chemotherapy or surgical decompression for children with large abdominal malignancies.4.3 For this reason, we adopted abdominal effusion for drainage and closed the abdominal wall primarily. This surgical strategy prevented postoperative bleeding that would be difficult to control.Because great risks exist for surgery for large abdominal malignancies in children complicated with ACS, the primary purpose of abdominal decompression is to reduce the abdominal pressure to save the child's life. Although a complete resection of the tumor through surgery is ideal, we believe that the complete removal of the tumor should not be blindly pursued. In this paper, for the first case of bilateral nephroblastoma complicated with ACS and the second case of abdominal alveolar rhabdomyosarcoma complicated with ACS, both had undergone 2 and 3 previous operations before the decompressive operation was necessary. Therefore, the first surgery was intended to remove a big part of tumor, using the irregular resection approach to effectively reverse the multiple organ dysfunction caused by ACS. After abdominal decompression, the children could undergo chemotherapy in a stable state, and the residual tumor shrank after chemotherapy, further providing conditions for a complete resection of the tumor. In the case of ACS occurring after the decompressive laparotomy, the delayed abdominal closure strategy is usually adopted.5In summary, abdominal decompression with surgical resection of the tumor is effective, and may be the only effective measure, to save children with large abdominal malignancies complicated by ACS.This study was supported by the Province Special Basic and Applied Basic Research Program ,The Guangzhou Collaborative Innovation of Industry, and the Major Projects of the People's Livelihood Project of Science and Technology (201604020151). This work was supported by the Key Laboratory of Malignant Tumor Molecular Mechanisms and Translational Medicine of Guangzhou Bureau of Science and Information Technology and the Key Laboratory of Malignant Tumor Gene Regulation and Target Therapy of Guangdong Higher Education Institutes.


Massive ovarian edema is a benign tumor like lesion of the ovary. The widely accepted mechanism is disruption of vascular drainage resulting in accumulation of fluid within the stroma and enlargement of the ovary. We report a case of massive ovarian edema in a teenage girl with hemoglobin SC disease. A 16-year-old female with hemoglobin SC disease was admitted with right lower quadrant pain. An ultrasound and CT scan showed a large, heterogeneous solid, and cystic pelvic mass. Due to the size and the possibility of malignancy, the patient underwent a salpingo-oophorectomy. The mass was an 8.3 cm hemorrhagic cyst with some solid areas. Histologic exam showed diffuse edema with scattered entrapped follicles and a narrow rim of normal appearing ovarian stroma. Dilated and occluded capillaries were seen along with hemorrhage and sickled red blood cells but no necrosis was identified. These histologic features were consistent with massive ovarian edema. Massive ovarian edema is thought to be caused by disturbance of the vascular outflow resulting in fluid buildup in the stroma. It is most often attributed to intermittent ovarian torsion that disrupts capillary and venous flow, but arterial flow is maintained. Rare cases of massive ovarian edema caused by tumor emboli or external compression by tumors have been reported, but this is the first case of a patient with hemoglobin SC disease developing vasoocclusions resulting in this lesion. Massive ovarian edema is benign tumor like lesion of the ovary caused by the accumulation of interstitial fluid resulting in ovarian enlargement. It is widely thought to be caused by the obstruction of lymphatics and veins, which results in the leakage of fluid out of the vessels. It is most often caused by external compression by neoplastic or polycystic ovaries with intravascular obstruction being less common. This is a unique case of massive ovarian edema caused by a vasoocclusive crisis in a girl with hemoglobin SC disease.A 16-year-old female was admitted to the hospital with a complaint of acute abdominal pain and vomiting. Her past medical history is significant for hemoglobin SC disease. She has had episodes of acute chest syndrome with the most recent in the fall of 2013. Her abdominal pain was described as sharp and was localized to the right lower quadrant with radiation on her right lower back. She states that although she has had abdominal pain caused by sickle cell crisis before, this current pain feels different. An ultrasound and CT scan was performed which showed a large, heterogeneous solid, and cystic pelvic mass. The differential diagnosis ranged from a teratoma to a germ cell tumor. Due to the size of the mass and the concern for malignancy, the patient underwent a salpingo-oophorectomy.A large, freely mobile dark purple mass with a translucent wall was seen originating from the right ovary. Upon attempt to remove the mass, a cyst ruptured, spilling straw colored fluid. After removal from the abdomen, the cyst was also found to contain clotted blood. No other abnormalities were seen within the abdomen or pelvis.Grossly, the mass was an 8.3 cm in greatest dimension diffusely hemorrhagic cyst with some solid areas. Cut surfaces showed diffusely hemorrhagic tissue with no recognizable ovarian tissue seen. Histologic exam showed extensive edema throughout the ovary. A narrow rim of ovarian stroma with luteinization was present along the outer surface . Rare foMassive ovarian edema is defined as the extreme enlargement of the ovary due to the accumulation of fluid within the interstitial space. The ovary has an extensive vascular network, most likely due to the fact that it is both a recipient and producer of hormones. If drainage within the lymphatic and/or venous component is disrupted then fluid will move out into the interstitial tissue . Since aOur case is unique in that it was caused by complications of hemoglobin SC disease, a condition that to our knowledge has not been previously reported. Hemoglobin SC disease is the second most common form of sickle cell disease, comprising about 30% of cases . It is dThe hypercoagulable conditions associated with hemoglobin SC disease along with the histologic features seen in the patient's ovary give clues to the pathogenesis.In this patient, the process most likely started within the small veins and lymphatics. The erythrocytes and neutrophils with their hypercoagulable properties began to adhere to the endothelium within these vessels. Eventually, these vessels became occluded, which increased the hydrostatic pressure resulting in fluid leaking out into the ovarian stroma. The disruption of the vascular drainage in the affected areas most likely caused release of inflammatory mediators, which then resulted in more vascular obstruction and more edema. The swelling then progressed until the ovary enlarged to the extent that it began to undergo torsion, which mechanically compromised arterial flow. This resulted in the symptomatic abdominal pain felt by the patient, which necessitated her presentation to the emergency room.Massive ovarian edema is enlargement of the ovary due to excessive interstitial fluid. It is caused by either intravascular obstruction or mechanical compression of lymphatics and small veins resulting in fluid extravasation. Hemoglobin SC disease is the second most common form of sickle cell disease. It is associated with hypercoagulative states, which can lead to vasoocclusive crises. This case is an example of a patient with hemoglobin SC disease developing primary massive ovarian edema caused by venous and capillary thrombi in a background of inflammation and hypercoagulation.


Isolated female epispadias without bladder exstrophy is a rare congenital anomaly affecting 1 in 484,000 females. The presenting features of female epispadias are urinary incontinence and abnormal anatomical features. A 6-year-old girl presented with primary urinary incontinence who on physical examination had a bifid clitoris and labia minora. The vagina and hymen were normal. Voiding cystourethrogram showed no reflux and a funnel shaped proximal urethra. With the diagnosis of isolated female epispadias, one-stage reconstruction of the urethra, bladder neck, labia minora and clitoris was performed. A 6-year-old female child presented with urinary incontinence and abnormal appearing genitalia since birth. The patient’s family also reported a history of recurrent urinary tract infections. The child was a product of nonconsanguineous marriage, born through a normal vaginal delivery and had normal developmental milestones. Inspection of external genitalia revealed a bifid clitoris and depressed mons . The labia minora was under-developed and terminated anteriorly to the corresponding half of the bifid clitoris. The central urethra lying above the vagina, was short and widely open dorsally, communicating with an open bladder neck that was incontinent of urine. Although routine biochemical parameters were within normal ranges, urinalysis revealed more than 13 red blood cells and 7–8 white blood cells per high power field. Urine culture grew Proteus mirabilis. There were no abnormalities on detected on intravenous urogram and ultrasound. Voiding cystourethrogram showed a small bladder capacity (70-80 ml) bladder with no vesicoureteral reflux . Pubic diastasis (width 2 cm) was noted. Urethrocystoscopy showed a short, wide urethra, approximately 1 cm long, with splaying of the roof of the urethra and a wide bladder neck. The ureteric orifices were situated 2 cm from the bladder neck and were of normal caliber. With the diagnosis of female epispadias, a combined Young-Dees and urethroplasty procedure was performed. The salient features included urethral plate tubularization, bladder neck and the proximal urethra relocation into the intra-abdominal position. The continence procedure was done by creating posterior strip of mucosa 15 mm wide and 30 mm long extending distally from the midtrigone to posterior urethra. The procedure was completed with cosmetic reconstruction of the external genitalia. The pubic symphysis was approximated. Suprapubic and uretheral tube were placed. Suprapubic tube was removed on 21st postoperative day, and uretheral tube on 10th postoperative day. At three month follow-up there was no incontinence of urine. Her mother reported that the child would express a sensation of a full bladder and a desire to micturate, with an ability to hold her urine and to initiate micturition voluntarily with complete bladder emptying. A postoperative voiding cystourethrogram after one year revealed a normal bladder neck and a lengthened urethra, with no reflux. A postoperative urodynamic study one year after the operation showed that the bladder capacity was 150 ml with a leak point pressure of 25 cm H2O. The postoperative cosmetic appearance was acceptable .Female epispadias is an uncommon congenital abnormality of the lower urogenital tract, which often presents with urinary incontinence, as in the index case. Usually it occurs sporadically but in some cases there is a strong genetic component.[1] The incontinence varies from continuous dribbling of urine without bladder filling to episodes of day-time stress-incontinence. Often the bladder capacity is reduced as a consequence of the lack of filling.[2] External genitalia can have varied appearance as classified by Davis ranging from lesser degrees with patulous urethral orifice to intermediate cases with urethra dorsally split along most of its length to the most severe cases which involve the entire length of urethra and bladder neck, rendering the sphincteric mechanism incompetent.[3] Milder forms of epispadias are extremely rare.[4] The objectives of surgical repair include achievement of urinary continence with preservation of the upper urinary tracts and the reconstruction of functional and cosmetically acceptable genitalia.[5] The same was achieved in the case presented. Source of Support: NilConflict of Interest: None declared


An osteoma is a benign tumor of bone with unknown etiology and is considered rare, mostly restricted to the craniofacial skeleton.This case report describes an uncommon condylar osteoma in a 67 years old white female patient with laterognathism to the left side, limited mouth opening, aesthetic change and pain associated with the right temporomandibular joint (TMJ). The histopathological examination confirmed osteoma. The lesion was surgically excised and immediate reconstruction was carried out using a custom-made total TMJ prosthesis. The patient has been in follow-up for 2 years, with no symptoms.Unilateral total TMJ prosthesis can be considered to replacement of TMJ after osteoma excision with resection of the condyle. An osteoma is defined as a benign tumor of bone resulting from the continuous formation of cortical and spongious bone. The etiology of the tumor is unknown, but may be associated with trauma, response to infections or inflammatory processes and growth abnormalities. Osteomas are rare and mostly restricted to the craniofacial skeleton. When they appear in the jaw region, there is a preference for the mandible rather than the maxilla. The most affected areas of the mandible are the body, angle and condyle –3.Osteomas in the mandible have been reported as a cause of trismus, limitation of mouth opening, progressive malocclusion with midline shift, contralateral mandibular deviation and facial asymmetry, especially when the mandibular condyle is involved. They may be symptomatic when their growth surpasses the limits of the bone –4. ReseaA total TMJ prosthesis must be designed so that it minimizes biomechanical stresses and at the same time achieves a homeostatic equilibrium –10. FiniThe custom-made total TMJ prosthesis “Promm” which is made in Brazil and is registered with ANVISA No. 10447390006 has been designed considering functional rehabilitation, anatomy and aesthetics of the patient.This study reports a case of an uncommon condylar osteoma that was identified due to an aesthetic change of the TMJ and immediate reconstruction using a total TMJ prosthesis.A 67-year-old white female patient was referred for the evaluation of asymmetry in the left lower jaw. On examination, the patient had laterognathism to the left side, limited mouth opening and pain in the right TMJ during maximum mouth opening and palpation. She reported that she had sought medical help due to extensive headaches. The doctor referred her to a dentist specialized in oral and maxillofacial surgery. The patient also reported the facial deviation that was becoming more and more noticeable to other people, interfering with her aesthetics Fig. a. The paFollowing the initial examination, a 3D CT scan was performed and it revealed an extensive but well-defined radiopaque lesion on the right condyle, measuring 1.5 by 2.0 cm . A computer numerical control (CNC) program used these images to machine the final model of the prosthesis. The mandibular component of the Arthroplasty System Promm consists of a condylar head made of cobalt–chromium–molybdenum alloy (ISO 5832-12) and a mandibular body made of pure titanium (ISO 5832-2), while the cranium component has a cavity where the condyle is articulated and machined in polyethylene of ultra-high molecular weight, UHMWPE (ISO 5834-2).All parts received a finishing process. The mandibular component was submitted to electro coloring, resulting in a blue color. All Promm products are batch mark, which allows the traceability of the raw materials the batch, provider, trader and date of each step, as well as the tools used for quality control of the parts.After performing asepsis, antisepsis and installation of drapes, the incisions were marked with a Skin Marker. At the location of the incisions lidocaine 2% with 1:50,000 epinephrine was infiltrated to improve homeostasis during the incision. The prosthetic joint was placed via the preauricular access and the submandibular incision access was used to fix the branch ramus and condyle. After exposure of the entire mandibular ramus, osteotomy was made with an incision safety margin until the trailing edge of the mandibular ramus, through the submandibular access, so that the condyle with the tumor could be removed via the preauricular access.The incised specimen was sent for histopathological examination, which confirmed osteoma. Figure After removal of the tumor, the prosthesis was adapted without difficulty and fixed in place with the 2.4 mm screw system. After implantation of the prosthesis the opening and closing mouth movements were tested and were found to be within normal limits. The patient has been in follow-up for 2 years, with no painful symptoms, mouth opening of 32 mm and she continues as an outpatient , and odontoma may be mentioned. Palatal and mandibular torus are exostoses which cannot be regarded as osteomas, although they are histologically identical , 15.Radiographically, osteomas show a well-circumscribed image with an oval or round radiopaque mass. Despite a radiographic image as described above, conclusive diagnosis can only be confirmed with a histopathologic examination , 4.Treatment for osteomas consists in complete surgical removal of the base containing the cortical bone. Recurrence is rare, but periodic clinical follow-ups are recommended as well as radiographic exams after surgical excision . Our patThe first case of a condylar osteoma was described by Ivy in 1927 . NowadayReconstruction of TMJ is a complex surgical procedure and it entails improved mandibular form and function, reduction of pain and disability, containment of excessive treatment and cost as well as and the prevention of further morbidity . The selIn conclusion, successful replacement of a TMJ with a custom-made total temporomandibular joint prosthesis after surgical removal of a benign tumor is possible. Unilateral total TMJ prosthesis can be considered as a replacement of a TMJ after osteoma excision with resection of the condyle.


This case highlights the need for clinical suspicion serial clinical assessment and radiological evaluation and the need for early surgery in patients presenting with abdominal symptoms following blunt abdominal trauma.Blunt abdominal trauma is a rare case of intestinal obstruction, and only few cases have been reported in the world literature. Stenotic intestinal obstruction following blunt abdominal trauma is a very rare complication Motor vehicle accidents have increasingly became the major cause of blunt abdominal injuries A 40‐year‐old male patient was brought to the Accident and Emergency with history of road traffic accident. Patient was examined and managed according to ATLS protocol.Pulse rate 88 per min, BP 104/84 mm of Hg, SPO2 98%.Abdomen soft with mild tenderness in both upper quadrants, FAST was positive with collection in the Morrison's pouch and perihepatic area.Polytrauma CT Scan: Brain – there was diffuse cerebral edema.Chest, abdomen, and pelvis including thoracic and lumbar spine: There was minimal hemopneumothorax on the right side. Large contusion involves the segments 7, 8, and 4 with minimal perihepatic collection. Contusion is along the superior border of spleen with minimum perisplenic collection. Fracture of transverse process of LI, L2.X‐Ray left leg: Comminuted fracture of mid shaft of left tibia and fibula.Blood Investigations: Hb 10.8 gm/dL, HCT 31%, platelet 129x10#3, glucose 114 mg/dL, creatinine .5 mg/dL, urea 17 mg/dL, sodium 139 mmol/L, alkaline phosphatase 310 U/L, ALAT 33 U/bilirubin 1.2 mg/dL, total protein 7.3 g/dL, globulin 3.1 g/dL.Patient was managed in the surgical ICU. After 2 weeks, he developed increased intra‐abdominal pressure, and laparoscopic drainage of 5 L of bile‐stained fluid was performed and drain was inserted. He underwent nailing of left tibia and discharged in good general condition after a follow‐up CT abdomen after 2 weeks.On 13.4.2015, patient presented to the Accident and Emergency with 5 days history of abdominal pain, constipation, and nausea. On physical examination, his vital signs were normal. Abdomen distended with hyperactive bowel sounds. Blood investigations including liver function test was normal. Plain X‐ray abdomen was showing distended small bowel loops with multiple air fluid levels. CT scan abdomen was showing dilated small bowel loops involving proximal jejunum and mid ileum with a zone of transition noted in the distal ileum suggestive of intestinal obstruction (Fig. Histopathology of resected segment of ileum with stenosis was showing marked inflammation, edema, vascular congestion, and features of perforation.Abdomen is the third most commonly injured body part following trauma and 85% cases are blunt abdominal trauma Blunt abdominal trauma is a rare cause of small bowel stenosis producing intestinal obstruction Common clinical symptoms of small bowel obstruction nausea, abdominal pain and vomiting or vague abdominal symptoms to frank obstruction Post‐traumatic small bowel stenosis is a clinical entity that needs to be watched for in all patients presenting with abdominal symptoms following blunt abdominal trauma managed conservatively.Written informed consent obtained from the patient for publication of this case report and accompanying images.SP: was primary surgeon in this surgery. HASB: assisted the surgery. OMAZ: assisted in laparoscopic drainage.None declared.