Lipofibromatosis is a rare fibro-fatty tumour with a predilection to involve distal extremities. It has only recently been described as a distinctive clinicopathologic entity, and subsequently only a few cases have been published in the literature. To address the clinicopathologic significance of this rare entity, we here describe a case of lipofibromatosis occurring on the left thigh of a Sri Lankan girl who developed a recurrence following excision.A 15-year-old previously healthy girl of Sri Lankan ethnicity presented with a painless progressively enlarging mass in her left thigh. Magnetic resonance imaging of her thigh lump, revealed a septated mass arising from subcutaneous tissue of anterolateral and medial aspects of her thigh. Histological assessment revealed evidence of lipofibromatosis, and the lesion was excised followed by split-skin grafting. She presented again with a local recurrence at the same site.Adequate surgical excision leads to complete cure of this benign lesion, but recurrences are common following incomplete excision. Therefore awareness among clinicians of this rare entity is vital in offering the best possible care to the patients. Lipofibromatosis is a recently described soft tissue tumour of obscure aetiology . We presA 15-year-old girl of Sri Lankan ethnicity presented with a painless progressively enlarging thigh lump for a period of 2 years. She was otherwise healthy. On examination a non-tender mass of soft consistency was noted arising from the anterior compartment of her thigh. There were no neurological and vascular compromises, and the lump was free of any deep attachments. Both hip and knee joints were clinically normal. Initial haematological investigations were within normal range.Following initial investigation, she underwent a magnetic resonance imaging (MRI) scan of her left thigh. A 6cm thick septate mass arising from subcutaneous tissue of anterolateral and medial aspects of her thigh was noted. Contrast enhancement following intravenous gadolinium was not noted. Underlying muscle and bone were normal Figures .et al. proposed that it should be classified as a separate entity as lipofibromatosis because it has distinct clinical and histological features [Lipofibromatosis is a rare benign neoplasm usually occurring in childhood. Although it was previously designated as infantile fibromatosis of non-desmoid type , Fetsch features .Lipofibromatosis commonly presents as a painless slow growing mass affecting distal extremities, such as hands or feet, and, as was the case in our patient, in proximal limbs, trunk or even head and neck regions -5. AetioOn histological examination the tumour is mainly composed of mature adipose tissue . AbundanUsually the mitotic rate is low and there is minimal cellular atypia . Unlike Diagnosis can be made with distinctive histopathologic features, although immunohistochemical studies are an invaluable aid in doubtful cases ,2. CommoImaging is an invaluable adjunct to diagnosing these rare tumours. On MRI, the detection of fat within the tumour is a valuable distinguishing feature between lipofibromatosis and the other soft tissue fibrous tumours . RadioloLipofibromatous hamartoma (macrodystrophia lipomatosa) of the nerves is a slow growing mass of fibro-fatty tissue surrounding and infiltrating major nerves. It is associated with macrodactyly and bone overgrowth .The clinical criteria for Proteus syndrome include the presence of a connective tissue nevus, ovarian cystadenomas or parotid adenomas as well as vascular malformations .Although it commonly presents as a painless slow growing mass, compression or entrapment of vessels, nerves and muscle has been reported previously with lipofibromatosis .Lipofibromatosis is notorious for local recurrence, as was the case with our patient. A high recurrence rate was noted following incomplete resection . CongeniIn conclusion, we propose that this rare and elusive clinical entity should be considered by clinicians and pathologists when encountering a soft tissue tumour in children and young adults. Although the number of documented cases is increasing, we believe that there is still insufficient clinical experience and lack of awareness about its existence. Since adequate surgical excision leads to complete cure of this benign lesion, awareness among clinicians is vital in offering the best possible care to the patients.Written informed consent was obtained from the patient’s legal guardian(s) for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.MRI: Magnetic resonance imaging.The authors declare that they have no competing interests.LS (registrar in surgery), TF and EL  gathered data. LS prepared the manuscript. HW (senior registrar in surgery) and KSP  made critical revisions to the original manuscript. LS, HW and KSP were responsible for overall surgical care of the patient. All authors read and approved the final manuscript.


Paratesticular liposarcomas are rare tumors that typically affect adult. Diagnosis is very difficult and inadequate surgical excision leads to a high rate of recurrence.We report a case of local recurrence of paratesticular liposarcoma diagnosed six months following surgery.Since there is low response to adjuvant treatments, extensive surgery remains the only curative approach, as shown by the case described here and the following review of the literature. Paratesticular liposarcomas (PLs), first reported in 1952, are rare tumors that comprise approximately 3 to 7% of all paratesticular sarcomas , 5. PreoIn this report, the authors report a case of local recurrence of PL diagnosed six months following inadequate surgical excision.A 50-year-old man, who had undergone right spermacelectomy in 2003, was admitted, in March 2011, to the General Surgery Department of the University of Roma ‘Sapienza’. He presented with a slowly growing mass located in his right hemiscrotum, with no other signs or symptoms. A physical examination demonstrated a right intrascrotal swelling, about 5 cm in maximum diameter, soft in consistency, with no pain and negative transillumination; the inguinal lymph nodes were nonpalpable. There were no specific abnormalities in the laboratory findings. An ultrasound (US) scan showed a 4.3 × 2.3 cm nonhomogeneous right scrotal lesion, diagnosed as a lipoma .The surgical specimen consisted of an irregular, seemingly circumscribed mass of yellow fat intermingled with a small amount of greyish, gelatinous zones.Microscopically, predominantly mature, adult-type fat cells were shown. Scattered among them were spindled atypical cells with enlarged, hyperchromatic, irregular-shaped nuclei and multivacuolated lipoblasts.Neither inflammatory cells nor dense fibrotic zones were found and a diagnosis of well- differentiated ‘lipoma-like’ liposarcoma was made.A monthly follow-up was carried out. Six months later, at the US scan of the right hemiscrotum, a nonhomogeneous 1.5 × 2 cm mass was revealed, with no evidence of tissue infiltration or pathological lymph nodes  according to World Health Organization (WHO) recommendations Figures  and 4.FiNo postoperative chemotherapy or radiotherapy was administered.At 24-month follow-up, the patient is doing well, with no clinical or radiological evidence of disease recurrence.PLs are rare tumors, 161 cases have been reported in the literature worldwide  and typiMost PLs are slow-growing tumors, with a huge palpable scrotal or inguinal mass (sized between 1.5 and 20 to– 30 cm in diameter), occasionally accompanied by slight to moderate pain and a sensation of heaviness. Due to this clinical presentation, differential diagnosis should include scrotal lipoma, groin hernia, hydrocele and epididymitis . Rapid gOn US scans, PLs are identified as heterogeneous solid, hypoechoic lesions with central colliquation due to necrosis, in the case of larger tumors. In smaller ones, US scans cannot always distinguish PLs from benign lipomas, as in the case we present. CT scans show the lower density of PLs compared to subcutaneous fatty tissue , 4.There is a correlation between tumor grade and histology and clinical behavior.well-differentiatedmyxoid and round cellpleomorphicde-differentiatedPLs are often classified in four subtypes:Most of these tumors (about 40 to- 50%) are well-differentiated (lipoma-like), sclerosing and inflammatory, according to current criteria , 3, 7. TThe term atypical lipomatous tumor embraces neoplasms with similar histological features but different clinical behavior, strongly influenced by location.Subcutaneous and intramuscular/intermuscular tumors do not metastasize and the rate of recurrence and dedifferentiation is very low, particularly in the former. In these cases, they are designated ‘atypical lipoma’.On the other hand, those located in central body sites are classified as ‘well-differentiated liposarcoma’ because they have high potential for recurrence and dedifferentiation. Although they can metastasize only when dedifferentiated, they can cause patient death simply by uncontrolled local growth.The differential diagnosis from lipoma, spindle cell lipoma and dedifferentiated liposarcoma is based on light microscopy examination because immunohistochemistry is of little value in distinguishing among these neoplasms.Implied in the foregoing statements, the most important role of the pathologist is to convey to the clinician the anticipated behavior of the lesion and the long-term risk of dedifferentiation.Difficulty in preoperative diagnosis of PLs can affect the correct surgical treatment. A correct preoperative diagnosis is infrequent, due also to the very low incidence of these tumors. For this reason, a surgical approach can be incorrect, or even incomplete. In these cases, the incidence of local recurrence is very high. The recurrence of well-differentiated PLs often involves dedifferentiated PLs, with worsening prognosis.Surgical treatment of PLs is radical orchiectomy and funicolectomy via the inguinal approach , with wide excision of involved local tissues.ab initio’. Relapse after a few months of a myxoid liposarcoma with areas of well-differentiated histotype cannot be explained otherwise. The histological misdiagnosis was made on the sample of the first surgical intervention. This may be attributed to one of the two followings factors: (1) sampling errors, myxoid areas were not identified and sampled for histological examination; (2) myxoid areas were a small part of the tumor and were not removed during the first surgical procedure.In our case, undoubtedly the tumor was a rare form of liposarcoma of mixed type ‘To date, due to the rarity of PLs, the role of radiotherapy and chemotherapy has not yet been well established , 7. The Paratesticular liposarcomas are rare tumors often located in paratesticular and spermatic cord tissues. When there is pre- or intraoperative diagnosis, radical orchiectomy and funicolectomy via the inguinal approach must be performed. In cases of incomplete surgery, we suggest performing an immediate radical procedure. The low response to adjuvant treatments imposes extensive surgery as the only curative approach, even in cases of smaller and well-differentiated tumors, due to the high risk of local recurrence in cases of nonradical excision.Written informed consent was obtained from the patient for publication of this Case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.


Dens invaginatus is a developmental anomaly that results in an enamel-lined cavity intruding into the crown or root before the mineralization phase. This report presents regenerative endodontic treatment of a necrotic immature tooth with Oehler's type III dens invaginatus of a nine-year-old female patient. A diagnosis of dens invaginatus (Oehler's type III) and a large periapical lesion was established with the aid of cone-beam computed tomography (CBCT). In the presented case contrary to the classic revascularization protocol, mechanical instrumentation was performed which apparently did not interfere with the regeneration process. After mechanical instrumentation of the invaginated canal by manual K-files, the invaginated canal space was disinfected by triple antibiotic paste followed by blood clot induction from the periapical tissues and the placement of mineral trioxide aggregate. At one-year follow-up, the tooth remained clinically asymptomatic. Radiographic examination revealed complete healing of the periapical lesion. At the 20-month follow-up, the radiographic examination also showed that the open apex was closed and the walls of the root canal were thickened. Dens invaginatus, known more commonly as “dens in dente,” is an unusual developmental anomaly resulting in a deepening or invagination of the enamel organ into the dental papilla before the mineralization phase. The etiology of dens invaginatus is still unclear and several theories have been suggested, including alterations in tissue pressure, trauma, infection, or local discrepancy in the cellular hyperplasia .In 1953, Hallet was the first to document an attempt to classify dens invaginatus. He suggested the existence of four types of invagination based on both clinical and radiographic criteria . A more The incidence of dens invaginatus has been reported to be in the range of 0.04%–10%, with the upper lateral incisors most commonly involved [Endodontic treatment of dens invaginatus type III can become complicated because of an unpredictable internal anatomy . A complIn the early 1960s, Ostby, a pioneer of regenerative endodontic procedures, demonstrated that new vascularized tissue could be induced in the apical third of the root canal of endodontically treated mature teeth with necrotic pulps and apical lesions. This was achieved by the creation of a blood clot in the apical third of a cleaned and disinfected root canal by using an apically extended root canal file just before root canal filling .In 2001, Iwaya et al. described a procedure which they termed revascularization. This was carried out on a necrotic immature mandibular second premolar with a chronic apical abscess. After a period of 30 months, they reported thickening of the root canal walls by mineralized tissue and continued root development .In 2004, Banchs and Trope described a revascularization protocol. After accessing the root canal, they irrigated it with sodium hypochlorite (NaOCl) and chlorhexidine gluconate (CHX) and sealed it in a combination of 3 antibiotics in an attempt to disinfect it and stimulate periapical repair .At present, the use of the term revascularization is debatable. Trope  claimed Lenzi and Trope  suggesteIn our perspective regeneration, the term indicates an overall goal of reproducing the original tissue histology and function and the current protocol should be revised to attain a more biocompatible strategy. More importantly, the effect of adding tissue engineering triad components  to the current protocol needs to be further researched . HopefulThe purpose of this report is to present a clinical case in which regenerative endodontic treatment was applied as a conservative method to successfully treat a maxillary lateral incisor with type III dens invaginatus and a large periapical lesion.A 9-year-old female patient was referred to Bakirkoy Acibadem Hospital with a complaint of pain on her maxillary right lateral incisor. She suffered from mild swelling associated with the tooth. Clinically, the crown of the maxillary right lateral incisor was cone-shaped and without caries. The tooth was sensitive to percussion. The periodontal status was normal (probing depth < 3 mm around the tooth). The tooth showed no mobility. The initial periapical radiographic examination revealed that the tooth showed an invagination (Oehlers' type III) and a large periapical lesion. The tooth had a diagnosis of symptomatic apical periodontitis. A CBCT scan was taken to see a three-dimensional image of this complex anatomy . The too2)  was then applied into the root canal.Regenerative endodontic treatment was recommended. After reviewing the risks, benefits, and treatment options with her parents, an informed assent and consent were obtained for performing the recommended procedure. Under local anaesthesia, rubber dam was placed and endodontic access was performed. A single canal orifice was exposed. The vital pulp tissue was seen in the invaginated canal but access to the main root canal was not possible. Because the invaginated canal was thin, it was instrumented up to a size 30 K-file (Dentsply-Maillefer) and irrigated with 2.5% sodium hypochlorite (NaOCl) solution. Calcium hydroxide (Ca(OH)Although the above-mentioned treatment was applied, the tooth was still symptomatic after three weeks. The buccal gingiva was swollen. So, as a further step, triantibiotic paste  was placed into the canal. One month after the antibiotic treatment, the tooth was asymptomatic.In the next phase of the treatment, the regeneration protocol was performed. Firstly, the triantibiotic paste was removed from the canal by irrigating the invaginated canal with 2.5% NaOCl and saline solution. Afterwards, the canal was dried with paper points. The irritation of the periapical tissues beyond the apex of the invaginated canal was performed using a size 30 K file. Five minutes after bleeding started, and a mineral trioxide aggregate (MTA) , plug was placed over the blood clot. A moist sterile cotton pellet was placed over the MTA and the access was sealed with a temporary restoration Cavit G . At the next appointment, two weeks later Fuji IX glass ionomer cement  and composite  were used in order to perform coronal restoration.The one-year follow-up showed no clinical signs of pathology. Radiographic examination revealed complete healing of the periapical lesion, but apical closure was not observed . The tooThe treatment of an immature permanent tooth with periapical pathosis is a challenge in paediatric dentistry, especially in the case of dens invaginatus. The various treatment options include apexification , nonsurg2 medication was not sufficient to eliminate the infection, so traditional apexification treatment was not suitable for the presented case.In the presented case, there was a large periapical lesion in the immature tooth with type III dens invaginatus. To promote root development, endodontic treatment should be performed. Apexification is the traditional treatment for necrotic teeth with open apices , but in Other nonsurgical endodontic treatment procedures are the complete elimination of the invagination with 70 or larger K files  or the rIn recent years, several case reports have showed that continued root development and complete periapical healing can be achieved with the treatment of an immature permanent tooth with pulp necrosis by a regenerative technique , 22. BecContrary to most of the published articles about classic revascularization protocol, in this case, mechanical instrumentation was performed in the invaginated canal to facilitate the insertion of intracanal medication, which apparently did not interfere with the regeneration process. In regeneration treatment, studies have reported different methods for disinfecting the necrotic immature tooth, including the use of triple antibiotic paste , 22 or cRadiographic follow-up after 20 months showed that the open apex had closed, while the walls of the root canal became thicker. Antibiotic paste and MTA caused the discoloration of the tooth . At thisThis report of pulp regeneration shows that the mechanical instrumentation of the invaginated canal in addition to intracanal medication with triple antibiotic paste led to satisfactory root development in a type III immature dens invaginatus with a large periapical lesion.


Although liposarcoma often metastasizes to various organs, cardiac metastasis, including to the pericardium, is rare. We present a case of a third recurrence of pericardial metastasis from the thigh, which required surgical resection because of cardiac failure. Surgery was effective for improving cardiac function and reintroducing chemotherapy. This is the first reported case of metastatic pericardial liposarcoma, which was successfully resected three times. We believe that aggressive surgical treatment, when it can resolve cardiac impairment, potentially leads to a more favorable prognosis. Liposarcoma is one of the most common soft tissue sarcomas in adults and often develops in the retroperitoneum and lower limbs. This tumor usually metastasizes to different organs, but metastasis to the heart, including to the pericardium, is rare. We report a patient with repeated cardiac metastases that required surgical resection.A 66-year-old woman was referred to our hospital because of recurrence of pericardial metastatic liposarcoma with progressive dyspnea. The patient had undergone surgical resection of a primary liposarcoma in the left thigh 19 years previously and resection of a locally recurrent tumor 13 years previously. She developed cardiac and pericardial metastasis 10 years later. Resection of a cardiac metastasis originating from the right ventricle was performed via median sternotomy 3 years previously, and a second resection followed by radiotherapy for recurrence of the pericardium metastasis adjacent to the diaphragm 2 years previously. Each of these metastatic tumors was large enough to result in cardiac failure. The pathological diagnosis of the resected primary and metastatic tumors was myxoid liposarcoma. The third recurrence of metastatic pericardial liposarcoma was detected 1 year previously and was followed by metastasis to the posterior mediastinum, liver, retroperitoneum, and peritoneum. Chemotherapy including gemcitabine, docetaxel, pazopanib, and epirubicin was administered for 1 year. Chemotherapy was effective for all of the metastases except those in the pericardium and posterior mediastinum. As the pericardial metastasis enlarged, the patient developed dyspnea over a period of several months. Her symptoms were speculated to be due to diastolic impairment by the large tumor. Reintroduction of chemotherapy was difficult because of cardiac failure. Therefore, she was referred to our hospital for surgical treatment of the pericardial metastasis.On admission, the patient had moderate dyspnea. Signs of cardiac failure, including engorgement of the jugular vein and edema of the lower extremities, were observed. Laboratory data showed an elevated brain natriuretic peptide level of 524 pg/ml. Chest X-ray showed a large mass lateral to the left ventricle extending to the left chest wall. Echocardiography showed a mass lateral to the main pulmonary artery in the pericardium. The left ventricular ejection fraction was preserved (60.9 %), but the inferior vena cava was dilated. Computed tomography showed a large, mostly low-density mass in the pericardium. This mass extended laterally to the ascending aorta and aortic arch, anterolaterally to the main pulmonary artery, and anteriorly to the left pulmonary artery Fig. . The masLiposarcoma is the second most common malignancy of soft tissues. The most common primary sites of liposarcoma are the retroperitoneum and lower limbs. Although liposarcoma often metastasizes to different organs, cardiac metastasis, including to the pericardium, is rare. Thirty-five cases of metastatic cardiac liposarcoma have been reported in the literature –6, with We successfully resected a pericardial metastatic liposarcoma, resulting in symptom improvement and reintroduction of chemotherapy. Aggressive surgical therapy may provide a symptom-free interval and prolonged prognosis.Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the editor in chief of this journal.


Development of infantile hypertrophic pyloric stenosis during postoperative period in EA with TEF is rare. Postoperative vomiting or feeding intolerance in EA is more common which is due to esophageal stricture, gastroesophageal reflux and esophageal dysmotility. A typical case of IHPS also presents with non-bilious projectile vomiting at around 3-4 weeks of life. The diagnosis of infantile hypertrophic pyloric stenosis in this subset is usually delayed because of its rarity. We report a case of IHPS in postoperative EA and emphasize on high index of suspicion to avoid any delay in diagnosis with its metabolic consequences.  A 2450gm baby boy was born to a primigravida mother at 39 weeks of gestational age at our institution through spontaneous vaginal delivery. The pregnancy was uneventful except detection of polyhydramnios at 28th week of gestation. Apgar score at 1st minute was 8. He had excessive salivation. The diagnosis of esophageal atresia (EA) was confirmed, as the orogastric tube could not be passed into the stomach. He was also noted to have malformed left thumb and anorectal malformation. Abdominal ultrasonography revealed absent left kidney. Echocardiogram showed a moderate sized patent ductus arteriosus (PDA) with left aortic arch. On his 2nd day of life, ligation-division of fistula and primary esophageal anastomosis performed via right thoracotomy; a sigmoid colostomy was also done. The baby remained ventilated and noticed to have saliva in chest drain on 5th postoperative day. The anastomotic leak healed on conservative management. The baby had stormy course during his NICU stay. He was treated for sepsis and fungal septicemia. The baby was discharged from hospital at D-31 when he was taking full oral feeds. In his follow up visit in outpatient clinic at 6 weeks after surgery, he was reported to have feed intolerance. A contrast esophagogram at 7th week revealed anastomotic stricture and reflux of contrast above the anastomosis. He was booked for esophagoscopy. In the meantime, a week later at the age of 8 weeks, he presented with large amount of milk-curd vomiting. An abdominal x-ray showed grossly dilated stomach. Diagnosis of infantile hypertrophic pyloric stenosis (IHPS) was suspected and was later confirmed on abdominal ultrasonography; pyloric muscle thickness measured 6.5 mm, pyloric diameter 13.6 mm and canal length 17.0 mm . An esophagoscopy done under general anesthesia and endoscopic dilatation confirmed an easy and pliable anastomosis. Ramstedt’s Pyloromyotomy was done in the same sitting. The feeds were started next morning. He was discharged home on 3rd postoperative day with the advice for regular follow-up. Unfortunately, the boy was readmitted at 5 months of life with diagnosis of aspiration and fulminant sepsis and he died within short period of time.Only 36 cases have been reported in the past literature [1]. The association of IHPS with EA was first reported in 1969 [2]. The absence of classical symptoms and signs of IHPS may be absent or masked after an EA or abdomino-thoracic surgery and this leads to delay in diagnosis. Diagnosis is usually confirmed after radio-imaging; upper Gastro-intestinal contrast study, ultrasonography or both . In our case, the baby boy had all components of VACTERL association except vertebral anomaly. He had minor anastomotic leakage which was treated conservatively. He developed anastomotic stricture which was initially attributed to his feeding problem. Moreover he had GER. Diagnosis of IHPS was done at 8 weeks of life. The strong suspicion of IHPS came after taking careful history of frequent episodes of milk curd vomiting noticed at 7th week and a dilated stomach on a plain x-ray abdomen. IHPS is so common that its occasional association with EA is probably coincidental. Symptoms of IHPS are easily attributable to the postoperative complication of primary surgery [5].In contrast to the common complications of EA surgery, IHPS does not come to consideration in postoperative vomiting. It is essential to be aware of increased incidence of IHPS in this group of patients. A high index of suspicion and diagnostic radio-imaging may make an earlier diagnosis and intervention.Source of Support: NilConflict of Interest:


A 66-year-old man with postsigmoidectomy status for colon cancer received laparoscopic partial hepatectomy due to a hepatic mass with employing titanium clips were for a vascular clamp. Histological examination showed liver metastasis from sigmoid colon cancer. Twenty-nine months after the partial hepatectomy, a mass developed on the stump at the hepatic resection. Laparoscopic left lateral segmentectomy was conducted under suspicion of cancer recurrence and an automatic titanium stapling device was used. The macroscopically cut surface of the liver showed a grey-white solid nodule measuring 23 x 20 mm and involving metal clips. The nodule was consistent with granuloma microscopically. Twenty-three months after the segmentectomy, a mass reappeared on the hepatic radial margin and an open left lateral hepatic lobectomy was performed because of its growth tendency. Histopathological examination revealed granuloma similar to the previous instance. Since these nodules formed a granulomatous lesion surrounding metal staples/clips and evidence of caseous necrosis was lacking, granuloma due to surgical staples/clips was suspected. Sporadic case reports of postoperative pulmonary granuloma at the staple line have been published previously, but there are no articles detailing a case involving hepatic granuloma. We present our case as the first report of postoperative staple-line hepatic granuloma. It is important for clinicians to distinguish liver tumors detected after surgical intervention of malignant disorders from recurrent or metastatic lesions originating from the primary cancer.Titanium is now widely used as a material in automatic stapling surgery because of its minimal irritability in the human body , but theA 66-year-old man underwent a colectomy for sigmoid colon cancer who had been initiated on hemodialysis for end-stage renal failure of unknown cause since 54 years of age and had no history of allergic diseases. Pathological examination of the cancer ultimately demonstrated a stage II type according to TNM classification. He had no sequential adjuvant chemotherapy. Fifty-eight months after the colectomy, a nodule was detected at the left hepatic lobe. A partial hepatectomy was then performed. Histological examination showed liver metastasis of the sigmoid colon cancer. Surgeons used metal clips to control bleeding at the cut surface of the liver.Twenty-nine months after the partial hepatectomy, complementary studies showed a mass lesion at the wedge from the cut stump at the hepatic resection. Computed tomography (CT) demonstrated a non-enhanced nodule measuring 35 mm in diameter Fig. . Gray-scMacroscopic examination revealed that the resected liver measuring 90 x 35 x 20 mm in size contained a grey-white solid nodule measuring 23 x 20 mm in maximum diameter. Microscopic examination confirmed that the nodule consisted of coagulation necrosis in the center Fig. . Silver The outermost layer was the band of granuloma comprising macrophages of epithelioid feature cells, lymphocytes and plasma cells with some of infiltration of eosinophils, which formed a distinct boundary between normal hepatic tissue and the necrotic region Fig. . MultinuNineteen months after the latest operation, a mass developed again in the resected edge of the liver . A contrast-enhanced CT of the abdomen detected a 45-mm low-density mass without contrast enhancement involving a linear, high-density material. An abdominal MRI scan indicated the mass had a low signal intensity on T1-weighted images and a slight high intensity on T2-weighted images. There was a strand-like component lacking signal intensity in the middle of the mass. MR diffusion-weighted imaging depicted the mass with a high signal intensity. The patient was not given a gadolinium-contrast agent because of his renal failure. Sonazoid®-enhanced ultrasonography revealed an avascular and hypoechoic mass at Segment 4 in the plain phase, and no contrast enhancement was observed at any phase. The FDG-PET/CT scan was not performed. Finally, at twenty-three months after the latest operation, open medial segmentectomy of the liver and caudate lobectomy was conducted under suspicion of cancer recurrence because of an increase of the mass in the short term. Operators did not use additional metal staples or clips in this procedure.Gross examination revealed a grey-white solid nodule measuring 45 x 38 mm in maximum diameter containing staples in the middle of the resected specimen Fig. . A two-dGranuloma possibly due to retained staples was suspected on the basis of the histopathological findings and clinical course of the patient. To confirm sensitivity to titanium or other metals, the patient was subjected to a patch test. Titanium, vanadium and aluminum were all negative. The patient did not show any obvious recurrence in the liver 16 months postoperatively.A search of the databases PubMed and Ichushi (in Japanese) using the query words “staple/stapler AND granuloma”, “clip/clipping AND granuloma”, “allergic granuloma” or “hepatic granuloma” yielded scattered reports involving the lung and granulomas possibly caused by surgical staples ,3, but tClips and staples applied for resection of the liver in our case were made of a titanium alloy . Titanium has been considered to be inert within the body , but sevAlthough the method generally used to examine metal hypersensitivity is the epicutaneous test , the patch test has been an examination procedure and mainly interprets the dermally-sensitized reaction and may not clearly reflect immune responses provoked by metal implants in deep tissue –16. Morein vitro examinations such as the lymphocyte transformation test (LTT) or its modified version, the memory lymphocyte immunostimulation assay (MELISA®) [Alternative testing methods have been utilized and include MELISA®) ,13,15. HMELISA®) ,13. Our Although there are several case reports of suspected hypersensitivity to titanium, the possibility remains of an allergic reaction induced by a small percentage of vanadium or aluminum released by corrosion of the titanium alloy . In the We report the first case of hepatic granuloma resulting from possible hypersensitivity to titanium alloy staples and/or clips. The three-layered structure consisting of necrosis containing titanium alloy, a desmoplastic (fibrous) area, and a band of granuloma could be interpreted as a histological characteristic for hepatic granuloma due to titanium alloy hypersensitivity.Written informed consent was obtained from the patient for publication of this Case Report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.


Intra-articular osteoid osteoma (OO) causes irreversible joint damage. Its treatment of choice is radiofrequency ablation (RFA); however, some areas of the acetabulum are hard to access. Therefore, hip arthroscopy was used to treat this tumor. We aim to systematically review the literature with regards to arthroscopic management of acetabular OO, and to report a further case in which hip arthroscopy was used for treatment.PubMed and EMBASE were searched for articles relevant to the arthroscopic management of acetabular OO on December 2, 2014. All articles published on and before that date were reviewed, and studies which met our pre-determined inclusion criteria were included. Articles screening and data abstraction were done by two reviewers independently. We also presented a 31-year-old man with acetabular OO who underwent hip arthroscopy for the management of his tumor after failing to respond to medications and computed tomography scan (CT)-guided RFA.The initial search revealed 14 studies, of which ten met our inclusion criteria. A total of ten patients underwent hip arthroscopy for the management of acetabular OO. Only two patients were females, and the patients' age ranged from 7 to 47 years. Two patients underwent arthroscopic guided-RFA of the lesion, while the rest underwent excision. The follow-up period ranged from 6 months to 2 years. Success rate was 100 %, and no recurrence was reported. Minor complications  developed in one patient (10 %). Arthroscopic-guided RFA failed to eliminate the tumor in our additional case. A second trial of CT-guided RFA was successful in treating the patient's condition.Hip arthroscopy is an effective and safe option for the management of acetabular OO, with success rate exceeding 90 %. Studies of higher level of evidence are required.The online version of this article (doi:10.1186/s12891-015-0779-8) contains supplementary material, which is available to authorized users. Osteoid osteoma (OO) is a solitary benign osteoblastic tumor, accounting for approximately 11-14 % of all benign bone tumors . PatientIntra-articular involvement of OO was observed in 10 % of the patients . When thOsteoid osteoma may spontaneously resolve after many years, or following prolonged use of NSAIDs , 14. OthOsteoid osteoma involves the acetabulum in around 0.5 % of the cases , 4. TreaTwo reviewers searched two online databases independently, PubMed and EMBASE, for papers related to arthroscopic treatment of acetabular OO published on or before December 2, 2014. The following subject headings and their related key terms were used: "osteoid osteoma", acetabulum", and "arthroscopy" . This included demographic information , clinical data , length of follow-up, patients lost to follow-up, arthroscopic technique used , treatment success, recurrence of the mass and complications. Success rate was considered as the primary outcome for this review. Due to the heterogeneity of the included studies, meta-analysis was not done, and a qualitative assessment is being presented. Individual study quality assessments were not performed as well because the included studies are of the level IV evidence.In addition to the systematic review, we present a 31 year-old male patient who underwent arthroscopic treatment for OO of the left acetabulum. Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the editor of this journal.The initial search revealed 21 studies. Out of these studies, seven duplicates were removed, and two articles were removed after title review. In addition, one article was excluded after abstract review since there was no available full text; the research was an abstract that was presented in a scientific meeting. Out of the 11 remaining articles, one article was removed after full text review because it described the use of arthroscopy for diagnostic purposes only. Eventually, ten studies were included in our review  failed to relieve his symptoms. Radiofrequency ablation therapy, consisting of three cycles for two minutes at 90 °C with an interval of one minute rest time between each cycle, was performed under CT guidance to treat the lesion. Following the procedure, the patient started to complain of severe pain that was limiting his ability to walk and stand normally even with continuation of NSAIDs. The OO appeared to be unchanged on a CT scan of the left hip that was done 4 months following the RFA. With the possible higher rate of complications of open surgery that requires hip dislocation, the patient agreed for an attempt of arthroscopic excision of the tumor. Based on the CT scans, the tumor was located posterior and superior within the acetabular fossa; therefore, hip arthroscopy was performed using the anterior, anterolateral and posterior portals. The region of the tumor was identified with irregularities of the cartilage at the posterior/superior edge of the fovea. Under fluoroscopic and direct guidance, a burr was placed in the identified region of the tumor. This region was burred, and the cavity of the nidus was identified. The burr could not be placed deeper inside the cavity due to its angulation, thus, the radiofrequency chisel was placed in the region and ablation involved the entire cavity. No intra- or post-operative complications were encountered.Six weeks after surgery, the patient reported no relief of his pain. A new CT scan of his left hip showed that the OO was still there, while the burned area appeared slightly posterior to the lesion. The patient underwent a second attempt of CT guided RFA of the lesion three weeks later. Improvement of his pain and increased range of motion of the hip were recorded two weeks following that. During the last follow-up visit, 44 months after the last procedure, he reported ignorable mild discomfort during activity. The nocturnal pain was gone, and he regained his ability to walk and stand without any pain. His range of motion improved fully with the exception of adduction at 10° and abduction at 30°. His latest follow-up x-rays showed no progression of the mild degenerative joint changes of the left hip which were noted in the first images Fig. .This article is the first to present a systematic review about the use of hip arthroscopy for the management of acetabular OO. Regardless of the technique used, hip arthroscopy was successful in all cases, and recurrence rate was 0 %. Minor complications  were reported in one (10.0 %) patient only. The use of hip arthroscopy failed to treat the tumor of our additional case, although the surgeon was an expert in performing such procedures. This emphasizes the difficulties the surgeon may face during arthroscopy for tumors in locations that are hard to reach. Although all areas of the hip can be visualized at the time of surgery, not all areas are amenable to arthroscopic resection. Resection of subchondral lesions requires removal of the overlying cartilage; this is not feasible in lesions in the major weight bearing areas of the acetabulum or femoral head. Moreover, even if a lesion is considered amenable to arthroscopic resection, there are instances in which the instruments’ design does not permit the surgeon to adequately address the lesion as described in our additional case. The difficulty in getting to the lesion because of the femoral head, acetabular location and instrumentation curvature resulted in the inability to burr deep into the subchondral bone and prevented a wider resection of the abnormal area. Our patient developed minimal cartilage/joint damage with the treatment provided; however, he got excellent relief of his pain and regained the range of motion of his joint eventually following the second trial of CT guided RFA.An intra-articular involvement of OO is rare and can result in misdiagnosis –5, 8. SyOpen surgical excision of OO has a high success rate that exceeds 88 % –33. In cComputed tomography-guided RFA represents a very successful modality of treatment of OO of the acetabulum resulting in rapid recovery and minimal complications , 17. SucAlthough this systematic review presents important data about the management of acetabular OO using hip arthroscopy, it has some limitations. The literature lacked studies of high quality and high level of evidence. Furthermore, a small number of patients were reported. Due to the fact that the acetabulum is a rare location for OO, researchers were not able to perform studies comparing open versus arthroscopic treatment for this tumor; all available studies were case reports. Moreover, inconsistencies of the surgical techniques used by the surgeons exist, and the tumors were present in different locations of the acetabulum among the patients. The duration of the post-operative follow-up of the patients was short; thus, late recurrence and degenerative changes to the joint could not be assessed. In addition, the results of hip arthroscopy could be affected by the surgeon's experience with this procedure. Although the success rate was high among the reported patients in this systematic review, surgeons who are planning to use a similar treatment option should be competent in performing hip arthroscopy .Hip arthroscopy appears to be an effective and safe option for the management of acetabular OO, with a success rate of more than 90 %. It should be considered when an image guided approach is not possible or likely to cause more joint damage. Nevertheless, location of the lesion in the acetabulum might limit proper resection. Studies of higher level of evidence are required.


Omphalopagus twins are conjoined twins sharing part of gastrointestinal system and abdominal wall. These types of twins have best chances of survival if successfully separated. We report a case of successfully separated omphalopagus twins at day six of life.  A 24-year-old multi-para underwent elective caesarian section in our hospital and delivered female omphalopagus twins . The combined birth weight of babies was 4.9 kg. The babies were designated as "twin A" and "twin B". The babies were fused from epigastrium to umbilicus and had a single umbilical cord. The bridge of tissue was firm, 2 cm thick and 5 cm in length. Both babies cried immediately after birth and passed urine and meconium. Ultrasonography of abdomen revealed fusion of liver and separate organ systems. Doppler ultrasound confirmed that the hepatic veins, portal vein and inferior vena cava of both twins were separate. Contrast CT scan showed differential enhancement of livers of both twins . The extra hepatic biliary system of both twins was separate with two gall bladders and common bile ducts. Rest of the organs were not shared. A 2-D echo study of twin A revealed 10 mm muscular VSD while twin B had small ASD with PDA. Blood investigations revealed Hb of 12.3 gm% for Twin A and 14.2 gm% for Twin B, with normal biochemistry values for both.Written informed consent was obtained from parents with the provision of saving the baby with better chance of survival if situation arises. The surgical plan, the anesthesia plan, location of anesthesia and surgical equipment, patient positioning and plan for repositioning after physical separation were discussed. Blood and blood products including fresh frozen plasma, platelets were reserved for surgery. Babies were taken for surgery on single operation table keeping one more table ready for closure of abdomen after separation. Separate anesthesia and surgery teams kept ready for each baby. Baby A was induced first followed by baby B. After anesthetic preparation was complete, the twins were lifted and surgically prepped on the posterior side first, placed on sterile sheets and then prepped anteriorly. Incision was taken on the connecting bridge of tissue. Skin and muscles were cut. Connecting part of liver was carefully divided using harmonic scalpel . Vital parameters of twin B who had minor cardiac anomaly showed slight deviation from normal but ultimately came back to normal by end of surgery. About 25 cc of packed red blood cells was transfused to each twin intraoperatively. After two and half hours of surgery, the twins were finally separated and twin B was taken to another table. Cut edges of liver of each baby were approximated with Vicryl 2-0. Abdominal drain placed in each baby and abdominal wall was closed. Both babies tolerated procedure well and extubated on table and shifted to pediatric surgery intensive care unit. Intravenous antibiotics were given till tenth post-op day. Oral feeds were started on post-op day four; abdominal drain was removed on post-op day five. Baby B had wound infection which was managed conservatively. Both babies were discharged on post-op day 12. Vaccination was done during hospital stay. Both babies have gained weight and on follow-up for five months.Conjoined twins prevail from 1:50,000 to 1:200,000; around 250 surgical separations have been reported so far. The largest series reported includes 17 sets of twins by Spitz et al. [1] and 25 sets by Cywes et al. [2]. The separation of conjoined twins presents a unique challenge to pediatric surgeons because of its rarity. Although omphalopagus twins have the best chances of survival, adequate team management and preoperative planning is required. Radiological investigations are needed for the evaluation of the shared organs, presence of anomalies, presence and extent of cross circulation.[3] The timing of surgery is controversial; however, a delay of few months gives better chances of survival. [1] Early separation is indicated when one twin threatens the life of the other.  Although major cardiac anomalies are contraindications of separations, variations in the cardiac functions have been observed with otherwise normal hearts as in our case.Conjoined biliary tract is reported in 25% of cases of omphalopagus twins. [5] The routine evaluation of cross circulation is performed using many methods like Tc-99m microcolloidal human serum albumin (HSA),Tc-99m HIDA, [6] injection of indigo carmine and the examination of its excretion in urine of the other twin. One method of evaluating cross circulation is contrast CT scan, [1] which in our case showed a watershed zone in the left side of liver bridge. Some have advocated glucose tolerance test for the evaluation of parasitism. [7] Intravenous fluorescein can be used to demarcate the large liver juncture. [7]Surgical separation should take the following into account: shared organs, soft tissue and bony defects that ensue after separation, age of the child and associated anomalies. Accurate preoperative investigations, a team approach, previous experience and meticulous operative and postoperative management contribute to the success rate. [6] After separation, there are often large areas devoid of skin; despite the use of preoperative tissue expansion, primary wound closure is not always possible, increasing the risk of postoperative sepsis in these individuals. [8] We could close the abdominal wound primarily in both babies though twin B had wound infection which required daily dressing. The long-term survival and outcome in relation to education and psychological outcome in separated twins is good. [9]Source of Support: NilConflict of Interest: Nil


Extraskeletal Ewing's sarcoma (EES) is a rare presentation, representing only 15% of all primary Ewing's sarcoma cases. Even more uncommon is EES presenting as a primary focus in the spinal canal. These rapidly growing tumors often present with focal neurological symptoms of myelopathy or radiculopathy. There are no classic characteristic imaging findings and thus the physician must keep a high index of clinical suspicion. Diagnosis can only be definitively made by histopathological studies. In this report, we discuss a primary cervical spine EES in a 53-year-old man who presented with a two-month history of left upper extremity pain and acute onset of weakness. Imaging revealed a cervical spinal canal mass. After undergoing cervical decompression, histopathological examination confirmed a diagnosis of Ewing's sarcoma. A literature search revealed fewer than 25 reported cases of primary cervical spine EES published in the past 15 years and only one report demonstrating this pathology in a patient older than 30 years of age (age = 38). Given the low incidence of this pathology presenting in this age group and the lack of treatment guidelines, each patient's plan should be considered on a case-by-case basis until further studies are performed to determine optimal evidence based treatment. Ewing's sarcoma is a malignant primitive neuroectodermal tumor (PNET) that primarily presents during the first two decades of life. Approximately 85% of those cases present primarily in the skeleton. With an annual incidence of 1–3 per million, it is generally a rare diagnosis . There hA 53-year-old male first presented to his primary care provider with a 2-month history of progressive complaints that started as a stiff neck and subjective right shoulder blade mass that was never objectively noted by any physician with some right upper extremity pain. This pain reportedly then spread to the left upper extremity with pain radiating down the posterior side to the left proximal wrist. Due to these complaints, he was evaluated by his primary care provider and started on ibuprofen. Upon awakening the following morning, the patient reported the sudden onset of left arm weakness that was not antigravity. Conversely however, the left upper extremity pain had remitted. With this sudden change in neurological function, the patient's primary care provider obtained an MRI of the cervical spine. The patient was told that the MRI showed a bony lesion and he was referred to our institution's orthopedics clinic. One month later he presented to the orthopedic clinic where the patient noted that his left upper extremity pain had returned. The patient and his wife also noted that, over the previous two-month period, he was losing muscle mass most profoundly in the left biceps and deltoid. Furthermore, he had also unintentionally lost 10 pounds over the previous three months. Due to his suspicious presentation and cervical spine lesion noted on the MRI, the patient was referred to our neurosurgical clinic.On examination, the patient appeared emaciated and easily fatigable. Muscle strength testing revealed 2/5 in the left upper extremity with exception of the biceps at 3/5 and handgrip of 4-/5. His right upper extremity was 3/5 in the deltoid and 4-/5 in all other muscle groups. The bilateral lower extremities were 5/5 for strength. There were no myelopathic signs. A repeat MRI performed at our institution showed an ill-defined predominantly extradural enhancement along the left aspect of the cervical spinal canal, most apparent at C6-C7, but also extending between C2 and C7 . A CT chThe patient underwent a C6-C7 laminotomy with decompression of the left C7 nerve root and biopsy of the extradural lesion, accomplishing the goals of operation including decompression and definitive diagnosis. Intraoperative baseline SSEPs were attempted; however no electrophysiological recordings could be obtained apart from minimal recordings in the right upper extremity.Histopathological studies established the diagnosis of Ewing's sarcoma. Our pathologists noted round, hyperchromatic nuclei with little cytoplasm and multiple mitosis suggestive of a primitive neuroectodermal tumor in surrounding necrotic tissue . FurtherUpon awakening postoperatively, the patient was noted to be severally quadriparetic and had suffered a myocardial infarction. He was transferred to the surgical intensive care unit where he was kept on flat bed-rest and high mean arterial pressure control. He recovered remarkably well. First, he regained movement in his lower extremities and subsequently regained antigravity ability in his right upper extremity. He continued to improve with the help of physical and occupational therapy. At his 6-month follow-up appointment his muscle strength was slowly improving with 3/5 in the left upper extremity with exception of handgrip, which was 4-/5. His right upper extremity was 4-/5 throughout. The bilateral lower extremities were 4/5 in strength. He was started on a course of chemotherapy and radiation therapy prior to discharge. To date, he has completed radiation therapy with a total treatment of 55.8 Gy. He is currently undergoing infusion treatment sessions of vincristine, actinomycin, and cyclophosphamide alternating with ifosfamide and etoposide.Extraskeletal Ewing's sarcoma of the spinal canal remains a rare diagnosis. It has been a subject of interest since its first published description in 1969 . EES affIn reviewing previously published case reports of primary EES of the cervical spine there were some patterns of presentation. The average age at presentation was 19.7 years and there were no cases identified that presented in a patient past 38 years of age. There was a slight propensity for the pathology to present in males (42%) compared to females (29%) in cases where gender was specified. However, one must take into account that 29% of the cases identified did not report the patient's gender. A further breakdown of these previous cases is summarized in Due to the rarity of this pathology, there are few large retrospective studies available for review. However, one such study performed by Ilaslan at the Mayo Clinic was able to identify 125 patients with primary spinal EES out of 1,277 patients with primary Ewing's sarcoma treated at the clinic from 1926 to 2001. The average age of the patients was 19.3 years (range 4–54 years). When the EES was organized by level in the spinal column, there were only a total of 4 patients identified (3%), further showing the infrequency of this pathology [In terms of imaging, reports vary and there appears to be no classical findings of an EES mass. On CT, there tends to be a mass that enhances heterogeneously . On MRI Definitive diagnosis is made by histopathological studies. Findings include uniform small round blue cells with scant possibly slightly eosinophilic cytoplasm notable on light microscopy. Two cellular markers frequently employed by pathologists are CD-99, which is expressed in both Ewing's sarcoma and lymphoma, and CD-45, which is only expressed in lymphoblastic lymphoma . TypicalConcerning treatment, there is strong evidence that early and aggressive treatment leads to the most favorable outcomes , 17, 23.Survivorship of primary EES of the spine post treatment was fairly consistent across different studies. A study by Venkateswaran from St. Jude's Children's Research Hospital of 33 patients showed an estimated 5-year survival rate of 48.1% (±8.9%) . This waThe occurrence of an extraskeletal Ewing's sarcoma remains rare, which typically affects adolescents with a male preponderance. It is especially rare to be found in the spine, especially the cervical spine. We have shown with this case report and in a review of prior literature that it is not unheard of for EES to present in the spine, regardless of age, and in our case, even in late adulthood. While it remains rare for an EES to present in the spine, it should remain a viable differential diagnosis of an ill-defined spinal mass noted on imaging, independent of the patient's age. Standard treatment often involves a multidisciplinary approach of surgery, chemotherapy, and radiation therapy with the particular preferential order determined on a case-by-case basis and has a survivorship of approximately 45% at 5 years after treatment.


MDM2 gene.Liposarcoma is the most common type of soft tissue sarcoma (STS). It is divided into five groups according to histological pattern: well-differentiated, myxoid, round cell, pleomorphic, and dedifferentiated. Dedifferentiated liposarcoma most commonly occurs in the retroperitoneum, while an intraperitoneal location is extremely rare. Only seven cases have been reported in literature. Many pathologists recognize that a large number of intra-abdominal poorly differentiated sarcomas are dedifferentiated liposarcomas. We report a case initially diagnosed as undifferentiated sarcoma that was reclassified as intraperitoneal dedifferentiated liposarcoma showing an amplification of the A 59-year-old woman with abdominal pain and constipation was referred to the Department of Advanced Biomedical Sciences, University of Naples Federico II, Naples, Italy, in November 2012. On physical examination, a very large firm mass was palpable in the meso-hypogastrium. Computed tomography (CT) scan showed a heterogeneous density mass (measuring 10 × 19 cm) that was contiguous with the mesentery and compressed the third part of the duodenum and jejunum.At laparotomy, a large mass occupying the entire abdomen was found, adhering to the first jejunal loop and involving the mesentery. Surgical removal of the tumor along with a jejunal resection was performed because the first jejunal loop was firmly attached to the tumor.in situ hybridization (FISH) analysis was performed and demonstrated an amplification of the MDM2 gene. Definitive diagnosis was intraperitoneal dedifferentiated liposarcoma.Macroscopic examination showed a solid, whitish, cerebroid, and myxoid mass, with variable hemorrhage and cystic degeneration, measuring 26 × 19 × 5 cm. Microscopic examination revealed two main different morphologic patterns: areas with spindle cells in a myxoid matrix and areas with pleomorphic cells. The case was initially diagnosed as undifferentiated pleomorphic sarcoma. Histological review showed areas of well-differentiated liposarcoma. Fluorescence No adjuvant therapy was given, but 5 months after laparotomy, the patient presented with a locoregional recurrence and chemotherapy with high-dose ifosfamide was started.No guidelines are available for the management of intraperitoneal dedifferentiated liposarcoma. We report this case to permit the collection of a larger number of cases to improve understanding and management of this tumor. Moreover, this study strongly suggests that poorly differentiated sarcomas should prompt extensive sampling to demonstrate a well-differentiated liposarcoma component and, if possible, FISH analysis. Soft tissue sarcoma (STS) is a rare mesenchymal tumor arising from non-epithelial connective tissue sources. The incidence of STS is approximately 4 to 5/100,000/year in Europe . LiposarDedifferentiated liposarcoma is a variant of liposarcoma with a worse prognosis and it most commonly occurs in the retroperitoneum, while an intraperitoneal location is extremely rare. Five cases of dedifferentiated liposarcoma in small bowel mesentery have been described . MoreoveMDM2) gene, in order to emphasize that in most cases of undifferentiated sarcomas, a specific line of differentiation can be demonstrated.We report herein a case initially diagnosed as undifferentiated sarcoma that was reclassified as intraperitoneal dedifferentiated liposarcoma showing an amplification of the murine double minute 2  and a positive test result for HBsAg. Tumor markers such as carcinoembryonic antigen (CEA), CA19-9, and CA-125 were within their normal ranges.Ultrasound revealed a very large mass with a complex echogenicity, almost completely occupying the lower quadrants of the abdomen. Computed tomography (CT) scan showed that the mass occupied the entire meso-hypogastric region, had heterogeneous density, involved the mesentery, and displaced the small bowel loops Figure . MoreoveAt laparotomy a very large mass was found with dense adhesions to the mesentery and sigmoid colon. The tumor seemed to arise from the first jejunal loop. A jejunal resection with mass excision and a gastrojejunostomy was performed Figure . We alsoMacroscopic examination of the specimen showed a very large, solid, whitish, cerebroid, and myxoid mass, with variable hemorrhage and cystic degeneration, a heterogeneous cut surface, and measuring approximately 26 × 19 × 5 cm. At an extremity there was a small intestinal resection of 10 cm that was on the serous surface of the tumor Figure A.Microscopic examination revealed two main morphologic patterns: first, areas that were less crowded with spindle cells in a myxoid matrix Figure B and occImmunohistochemical stains for cytokeratin, CD34, S100, actin, calretinin, and CD117 (c-Kit) were negative, while Bcl-2, EMA, and CD99 were positive in the neoplastic cells.The histopathological diagnosis was of an undifferentiated pleomorphic sarcoma.in situ hybridization (FISH)analysis was performed and demonstrated an amplification of the MDM2 gene. These findings were consistent with an intraperitoneal dedifferentiated liposarcoma.Histological review showed areas of well-differentiated liposarcoma and the tumor was reclassified as dedifferentiated liposarcoma. Moreover, a fluorescence Intraperitoneal dedifferentiated liposarcomas are rare and, to our knowledge, only eight cases have been reported to date Table . Of thesAWD, alive with disease; DOD, died of disease; NED, no evidence of disease; RTX, radiation therapy.Imaging modalities may help in preoperative diagnosis of the origin of tumor. However, CT findings may suggest the diagnosis of liposarcoma when the tumor contains areas of fat attenuation, which is less likely to be seen in higher-grade liposarcomas. Moreover, dedifferentiated liposarcomas demonstrate marked heterogeneity on magnetic resonance imaging (MRI), with areas of necrosis and heterogeneous contrast enhancement, indistinguishable from other high-grade sarcomas -15.Accurate diagnosis demands an experienced pathologist, the use of immunohistochemistry, and often also cytogenetics ,17. MicrMDM2[MDM2 amplification by immunohistochemistry, FISH, quantitative PCR, or comparative genomic hybridization (CGH) may prove an adjunctive tool in the diagnosis of lipomatous neoplasms. This feature can be of remarkable help in reaching the diagnosis of dedifferentiated liposarcoma, particularly in cases of poorly differentiated sarcomas for which a specific line of differentiation cannot be demonstrated, as observed in this case.Well-differentiated liposarcomas/atypical lipomatous tumors and dedifferentiated liposarcomas have been cytogenetically shown to harbor ring and giant marker chromosomes consisting of amplicons in the 12q13-15 region, resulting in amplification of several genes, including most notably MDM2,20. IdenRadical excision of the tumor offers the possibility of longer survival and a disease-free interval. In all patients reviewed in the literature, surgical excision was the first line of treatment of intraperitoneal liposarcoma. The value of adjuvant chemotherapy has not been established. Postoperative radiotherapy to the whole tumor bed is not feasible at a tolerable toxicity ,22. One In terms of recurrence, follow-up information is available for all cases reported. Only one patient did not have recurrence. Of the eight patients, three died of disease.MDM2 amplification using FISH are recommended.Surgery is the main treatment and the role of adjuvant therapy is widely debated.In order to improve overall understanding of the intraperitoneal liposarcoma, it is useful to analyze the collected data of all observed cases.In summary, the rarity of intraperitoneal dedifferentiated liposarcoma makes it difficult to investigate clinical characteristics, therapy, and outcomes. Differential diagnosis of this tumor can be very difficult because the histological examination does not demonstrate a specific line of differentiation. Therefore, in cases initially diagnosed as poorly differentiated sarcomas, extensive sampling and an analysis of Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.in situ hybridization; MDM2: Murine double minute 2; MRI: Magnetic resonance imaging; PCR: Polymerase chain reaction; STS: Soft tissue sarcoma.CEA: Carcinoembryonic antigen; CGH: Comparative genomic hybridization; CT: Computed tomography; FISH: Fluorescence The authors declare that they have no competing interests.CG carried out acquisition, analysis and interpretation of data, and drafted the manuscript. AC and NC critically revised the study. SC made substantial contributions to acquisition, analysis, and interpretation of histopathological data. FPD carried out histopathological examination and critically revised the study. AR performed the surgical operation, conceived the study, and participated in its design and coordination. All authors read and approved the final manuscript.CG: specializzando in Chirurgia Generale, Dipartimento di Scienze Biomediche Avanzate, Università Federico II, Naples, Italy; AC: specialista in Chirurgia dell’ apparato digerente ed endoscopia digestiva chirurgica, Dipartimento di Scienze Biomediche Avanzate, Università Federico II, Naples, Italy; NC: dirigente medico, Dipartimento di Scienze Biomediche Avanzate, Università Federico II, Naples, Italy; SC: specializzando in Anatomia Patologica, Dipartimento di Scienze Biomediche Avanzate, Università Federico II, Naples, Italy; FPD: professore ordinario di Anatomia Patologica, Dipartimento di Scienze Biomediche Avanzate, Università Federico II, Naples, Italy; and AR:professore ordinario di Chirurgia Generale, Dipartimento di Scienze Biomediche Avanzate, Università Federico II, Naples, Italy.


Symmetric lipomatosis is definitively characterized by symmetric, tumorous lipomatous proliferation of adipose tissue that often develops in the head and neck, shoulders, and upper trunk. However, in the oral region, symmetric lipomatosis of the tongue (SLT) is an extremely rare condition related to generalized lipidosis that is often caused by chronic alcoholism. It is characterized by multiple symmetric lipomatous nodules and diffuse bilateral swelling located within the tongue. We report an extremely rare case of SLT arising in an 80-year-old man with a long history of alcoholic liver cirrhosis. He exhibited multiple soft nodular protrusions on the bilateral margin of the tongue presenting as macroglossia for years. Although MR imaging showed multiple fatty masses on both sides of the tongue, there was no elevated tumor mass on the bilateral margin. The patient underwent bilateral partial glossectomy under general anesthesia. Histopathologically, the resected tumor exhibited diffuse infiltration with mature adipose tissue lacking a fibrous capsule. Due to the lipidosis and the unusual presentation of multiple lesions, the lesion was ultimately diagnosed as SLT. At present, after surgery, the patient wears a full-denture and is in excellent condition, with no sign of recurrence, improved QOL, and recovery of masticatory, articulatory, and speech intelligibility functions.  Benign symmetric lipomatosis (BSL) was first reported by Brodie in 1846  in 1944,2). Extraoral findings showed facial symmetry and a good complexion. There were no significant findings in the cervical lymph nodes. Preoperative laboratory examination revealed high levels of TG (202 mg/dL), but TC and HDL and LDL-cholesterol were normal.The patient was an 80-year-old man who was referred to our division for the chief complaints of macroglossia and articulatory disorder. Oral findings showed painless tumors on the bilateral margins of the tongue and dysarthria/dysphasia . His medMultilocular tumors were found on the bilateral margins of the tongue, which formed macroglossia, and xanthic, elastic soft tumors were evident in the shallow submucosa. The surfaces of the tumors were smooth, and in particular, the left tongue margin was significantly elevated.While there were no abnormalities in perception/taste, dysarthria was observed. MR images showed T2 hyperintensity and well-defined borders in the bilateral margins of the tongue and ill-defined trabecular structures like muscle bundles in the deep part of the tongue, which suggested that it might be symmetric lipomatosis . The patThe resected specimens showed a relatively well-defined border in the mucosa but the encapsulation was not clear in the deep portion .The tumor consisted of proliferation of mature adipocytes, and there were no signs of encapsulation in the muscle layer or atypia, such as mitotic figures Figures . Based oAt the present time, there has been no sign of postoperative dysfunction or complication, and the dysarthria and dysphasia have improved significantly. The patient's clinical course remains excellent .Lipoma is the most common nonepithelial benign soft tissue neoplasm and monostotic solitary lesion and can occur in any part of the body. Meanwhile lipomatosis is one of multiple systemic tumors that are characterized by diffuse proliferation of adipose tissue in normal tissues. Lipomatosis is differentiated from lipoma by specific characteristics, such as ill-defined borders and nonencapsulation. However, while lipomatosis is relatively rare in the oral region, it can sometimes develop bilaterally on the tongue.Lipomatosis in the tongue is classified by gross appearance into two types, nodular type and diffuse type . The nodWith respect to the association between SLT and alcoholic liver injury, fatty deposits in the body might occur due to increased blood acetyl CoA levels, which is the primary source of fatty acid derivatives during the metabolism of alcohol to acetate by alcohol dehydrogenase and acetaldehyde dehydrogenase in the liver , 10.The most common causes are alcohol dyslipidemia and liver damage, as in the present case, followed by HT, viral hepatitis, and stroke. The local factors include fatty degeneration with aging of the superficial lingual muscle, hyperplasia of the fat layer of the lingual septum, adipose tissue formation during granulation formation caused by occlusion error, and stimulation and trauma caused by the teeth/occlusion. In our case, we considered that the systemic factor was alcohol dyslipidemia and that the local factors were fatty degeneration with aging of the superficial lingual muscle, which was proposed by Staudt et al. , and hypRecently, there have been some reports of treatment strategies, such as tumor resection and surgical debulking, with consideration also given to prevention of dysfunction . Recentl


Ossifying tendinitis (OT) is a type of heterotopic ossification, characterized by deposition of hydroxyapatite crystals in a histologic pattern of mature lamellar bone. It is usually associated with surgical intervention or trauma and is more commonly seen in Achilles or distal biceps tendons, and also in the gluteus maximus tendon. To our knowledge, there is no description of OT as a complication of calcifying tendinitis of the rotator cuff. In this report, we describe two cases in which the patients developed an OT of the supraspinatus after arthroscopic removal of calcium deposits. The related literature is reviewed. Subacromial calcium deposits and calcifications in the tendons of the rotator cuff (RC), with histologic presence of chondrocytes along tenocytes, were identified as a cause of scapulohumeral periarthritis in the early 1900s . Later, Ossifying tendinitis (OT) is a type of heterotopic ossification (HO), characterized by deposition of hydroxyapatite crystals in a histologic pattern of mature lamellar bone , 8.It is usually associated with surgical intervention or trauma  and is mIn April 2005 the patient came to our outpatient office, complaining of severe pain and discomfort in the right shoulder for 1 year. After radiological and ultrasound (US) examination, he was diagnosed with calcific tendinitis of the rotator cuff and he underwent two cycles of extracorporeal shock wave (ESW) therapy. At 1 year follow-up he had not had any improvement in pain and shoulder function and therefore was advised to undergo shoulder arthroscopy.Preoperative shoulder examination showed the following range of motion (ROM): 160° in flexion and abduction, 80° in internal rotation(IR) and 90° in external rotation(ER). Impingement tests (Hawkins and cross-arm) and Empty Can Test were found positive. The Constant–Murley score (CS) was 67 points, and the Simple Shoulder Test (SST) had a 5/12 “yes” response. Laboratory exams showed normal values of peripheral blood counts, and X-ray showed a 1.2-cm calcification located on the bursal side of the supraspinatus tendon  failed and he was therefore prescribed shoulder MRI that revealed slight changes in signal intensity (T1 weighted) of the supraspinatus insertion due to degenerative alterations of the tendon Fig. d. A thirThe patient followed the standard postoperative program and he had slight pain for 3 years, especially during work activity. At the last FU examination in December 2013 (8 years) the CS was 87 and SST had 10/12 “yes” responses.CT of the shoulder is a widespread clinical condition with a significant impact on patient’s quality of life. Although several treatments have been proposed, the best option to choose is still controversial –16. ExtrIn a case series of 892 patients treated with acromioplasty and distal clavicle resection, Berg et al.  reportedHO of the deltoid muscle  and suprThe mechanism of origin of bone metaplasia in the RC tendon with calcium deposits is unknown, but some aspects of this phenomenon can be interpreted through the findings already known to us. The presence of resident progenitor cells with multi-differentiation potential in the human tendon  and locaIn both cases the RC was involved with severe pain and functional impairment that required an arthroscopic second look to ascertain the origin and the characteristics of the mass.During the surgical procedure of case 1 we found a formation of hard consistency above and partly within the supraspinatus tendon at the same site the first calcific deposit was removed from; in case 2 we found similar macroscopic characteristics of the ossification, with a tendency to infiltrate the tendon. The histologic features showed in both cases areas of chondrometaplasia and ossification that were diagnosed as a particular form of OT, without supposing such a kind of evolution before the intraoperative assessment.The ossifications found above and within the substance of the tendon may be the result of a transformation of mesenchymal cells to bone-forming cells in response to the surgical excision of the calcium deposit and suturing of the tendon during the arthroscopic procedure.Our preference for complete, meticulous excision of the mass might help avoid further recurrence of the ossifying mass. The long-term follow-up of the two cases described in this study showed no clinical or radiological recurrence of the deposits. Although the surgical approach may have been the trigger event inducing the chondrometaplasia, we have not enough data to support this speculative hypothesis, nor can we rule out that the ossification and cartilaginous metaplasia could be the natural evolution of the case. The surgical findings described in this study led us to consider with caution arthroscopic excision of calcium deposits and to be meticulous during the subacromial debridement of calcific foci to minimize the risk of recurrence. We do believe that the description of these two rare cases of OT will be useful to include this condition as a further complication of CT and also to consider the shoulder as an additional potential site of OT.


Small bowel malignant tumors are rare and sarcomatoid carcinomas have rarely been reported at this site. We report a 56-year-old woman, with history of an excised gliosarcoma, who presented with recurrent obscure gastrointestinal bleeding. She underwent endoscopy and colonoscopy, which failed to identify the cause of the bleeding. The abdominal computed tomography scan located a tumor in the small bowel. Pathology revealed a jejunal sarcomatoid carcinoma. She developed tumor recurrence and multiple liver metastases shortly after surgery. Immunohistochemistry is required for accurate diagnosis. Sarcomatoid carcinoma is a rare cause of obscure gastrointestinal bleeding, which is associated with a poor prognosis. The American Gastroenterological Association defines obscure gastrointestinal bleeding (OGIB) as bleeding of unknown origin that persists or recurs without an obvious etiology after a normal endoscopy (colonoscopy and upper endoscopy). It can be categorized into overt gastrointestinal (GI) bleeding with recurrent melena or hematochezia and occult GI bleeding with iron deficiency anemia and/or positive result on fecal blood test , 2.OGIB accounts for approximately 5% of patients with GI bleeding. In approximately 75% of these patients, the source is in the small bowel, which is difficult to assess with conventional endoscopic and radiological procedures , 3. EvalThe introduction of CE and DBE and the recent improvements in computed tomography (CT) and magnetic resonance imaging (MRI) techniques have revolutionized the approach to patients with OGIB, allowing the visualization of the entire GI tract, particularly the small bowel—until now considered as the ‘dark continent’ . We descA 56-year-old woman with a past history of a right temporal gliosarcoma  was admitted on two occasions in May 2012, with asthenia and two previous episodes of coffee ground vomiting. An initial upper endoscopy showed no signs of active bleeding. One month later, she was re-admitted due to persistent anemia and a 3–4 kg weight loss. On physical examination, she was pale. Rectal examination was normal. Laboratory results showed: hematocrit, 18.3%; hemoglobin, 5.7 g/dL; serum iron, 10 μg/dL (37–45); erythrocyte sedimentation rate, 131 mm/h; c-reactive protein, 143 mg/L; AST, 103 U/L; ALT, 87 U/L; gamma-GT, 189 U/L; alkaline phosphatase, 573 U/L; total bilirubin, 0.6 mg/dL. The rest of the laboratory tests, including tumor markers, were normal.A colonoscopy was performed but showed no abnormal findings. An abdominal ultrasound showed a solid C-shaped mass, 9 cm in diameter, at the level of the pelvis, probably arising from the small bowel. A subsequent abdominal/pelvic CT scan  showed aMacroscopic examination of the specimen revealed a 10 × 6.5 cm small bowel mass that was extensively ulcerated and had areas of necrosis. Microscopically, the tumor showed sheets of pleomorphic spindle cells arranged in fascicles with vesicular nuclei, small nucleoli, and eosinophilic cytoplasm. There was no evidence of epithelial differentiation. Mitotic figures averaged 35–40 per 10 high-power fields. The surgical margins were free of involvement. The tumor extended through the wall of the jejunum into the surrounding tissue and extensively invaded the serosa. Eleven lymph nodes were dissected from the mesentery, three of them showing presence of tumor deposits (TNM stage pT3 pN1 M0). Immunohistochemistry revealed strong positive staining for vimentin and cytokeratin (AE1/AE3 and CAM 5.2). Epithelial membrane antigen (EMA) was focal positive. Other markers were negative (CD 117 (c-Kit), S-100 protein, DOG 1, CD 34, smooth muscle actin (SMA), caldesmon and desmin. The diagnosis of jejunal SCA was made .Figure Adjuvant chemotherapy was planned following surgery. Two months later the patient developed multiple liver metastases. After four months of regular follow-up, left-sided hemiparesis recurred and MRI revealed tumor recurrence in the right temporal lobe. A body CT scan showed a recurrent small bowel mass, with abdominal disease progression. She died six months after diagnosis.The etiology of OGIB is strictly age-dependent, with angiodysplasia and drug-induced small bowel injury being most common in patients more than 40 years of age. Among patients younger than 40 years, small bowel tumors, Crohn’s disease, Meckel diverticulum, and polyposis syndromes are the most likely causes , 3. AngiSmall bowel tumors are the second most common cause of OGIB, accounting for 5–10% of cases. Several different types of tumor can arise within the small bowel, both malignant , and benign  , 5, 6. SNon-steroidal anti-inflammatory drugs are the most frequent cause of drug-induced small bowel injury and the damage mainly occurs in the ileocecal region. The injury, even when not severe, can cause persistent bleeding and iron deficiency anemia. Generally, there is 2–10 mL of daily blood loss. Frank, acute GI bleeding is relatively rare and is caused by ulcers and erosions . Other lOur patient had a gliosarcoma, which is a rare primary tumor of the brain, classified by the World Health Organization as a high-grade glioma and a variant of glioblastoma multiforme, consisting of both gliomatous and sarcomatous components. Multiple reports in the literature describe the greater tendency of gliosarcoma to metastasize to extracranial  and intra-axial sites . There are reported cases of metastases to skin, chest wall, pleura, spleen, kidneys, bone marrow, adrenal and thyroid gland, pericardium, myocardium, diaphragm, stomach and pancreas. Gliosarcoma has a poor prognosis, due mainly to high recurrence rates . In our SCA is a rare variant of small intestinal carcinoma, which displays both carcinomatous and sarcomatous features , 11. GasBecause monophasic SCA can be confused with other sarcomas due to the absence of carcinomatous features, a wide-range panel of immunohistochemical biomarkers should be performed for the differential diagnosis . LeiomyoSmall bowel carcinomas have a poor prognosis, but that for SCA is far worse, because they are highly aggressive tumors and most patients present with advanced disease . Our patAfter a search in PubMed using the words, ‘sarcomatoid carcinoma’ and ‘gliosarcoma’, we have not, to date, found any previously published case associating both tumors, so we consider that this association was coincidental in our patient. We cannot exclude the possibility that the patient had an unrecognized cancer predisposition syndrome  given the fact that she developed two rare types of tumor, and her father died of lung cancer at the age of 58.In summary, both gliosarcoma and SCA are very rare tumors with high tendency to metastasize because of the sarcomatous component, resulting in a high mortality and recurrence rates. SCA of the small intestine must be kept in mind in the management of obscure GI bleeding.Conflict of interest: none declared.


Neurofibromatosis type 1 (NF1) is a genetic disorder that disturbs cell growth in nervous system, causing tumors to form on nerve tissue. A 26-year-old man was brought to Ali-Ebn Abitaleb Hospital, Rafsanjan, Iran, with asymmetric quadriplegia more in left side. He was a known case of NF1. He first had a paraparesia two weeks before admission that progressed to quadriparesia. There was no family history of neurofibromatosis.Hyperpigment lesions were revealed in variable sizes. Multiple nodular lesions and inguinal freckles were seen on his body surface. Babinski sign, hyperreflexia and spasticity were detected in lower limbs examination. Decreased sensation was revealed more in left side. Sphincter function was normal. In cervical spine magnetic resonance imaging (MRI), there were multiple mass lesions in paraspinal, intradural extramedullary and extradural region with cord compression from C1 to C6. Several neurofibromas were extraordinary in orbital cavity, neck, whole paraspinal tissues, mediastinum, para-aortic region, presacral, and pelvis with widening of neural foramina (dumbbell shaped lesions), also in perianal and sacral region -3.Three months later, he was admitted to the surgery ward for an extensive laminectomy and reconstruction procedures.1As the most common symptom in other reports, our presenting case symptom was quadriparesis but asymmetric. In our case, there was multi-level involvement of cervical cord, not only C2 or C3 as in most reported cases.1 In another case report, a 43-year-old man with history of NF1 presented with signs and symptoms of myelopathy and severe cervical kyphotic deformity.2 Our case was unique according to absence of deformity with widespread and plexiform neurofibromas.In a retrospective study on 97 patients with NF1, spinal curvature abnormality was present in 50/97 patients.3In literature, there were some reported extensive neurofibromas that were less widespread than ours; and also there were 9 reported cases between the age of 3 to 15 years old with voluminous plexiform neurofibromas in neck region that were not as widespread as our case.Our case had diffuse involvement from orbital to sacral region and such extent of involvement has not been reported in related articles about patients with neurofibromatosis.


Odontogenic cutaneous sinus tracts are often misdiagnosed as lesions of non-odontogenic origin, leading to the treatment of patients with unnecessary and ineffective therapies. Sinus tracts of endodontic origin usually respond well to endodontic therapy. However, root canal treatment of mandibular molars with aberrant canal anatomy can be diagnostically and technically challenging. Herein we present a patient with a cutaneous odontogenic sinus tract in the right submandibular area.A 23-year-old Chinese female patient presented with a cutaneous odontogenic sinus tract that was initially misdiagnosed as a sebaceous cyst. The patient had undergone surgical excision and traditional Chinese medical therapy before endodontic consultation. With the aid of cone beam computed tomography (CBCT), it was confirmed that the causative factor of the cutaneous odontogenic sinus tract was chronic periapical periodontitis of the right mandibular second molar, which had a rare and curved distolingual root. The resolution of the sinus tract and apical healing was accomplished following nonsurgical root canal treatment.A dental aetiology must be included in the differential diagnosis of cutaneous sinus tracts in the neck and face. Elimination of odontogenic cutaneous sinus tract infection by endodontic therapy results in resolution of the sinus tract without surgical excision or systemic antibiotic therapy. This case report also indicates that CBCT imaging is useful for identifying the tooth involved, ascertaining the extent of surrounding bone destruction and accurately managing the aberrant canal morphology. Odontogenic cutaneous sinus tracts are rare dermatoses that occur because of chronic dental draining infections, especially apical periodontitis ,2. The mThis case report presents an odontogenic cutaneous sinus tract that was initially misdiagnosed as a non-odontogenic lesion. With the aid of cone beam computed tomography (CBCT), we identified the cause to be the right mandibular second molar with a rare and severe curved distolingual root.A 23-year-old Chinese female patient was referred to the Department of Conservative Dentistry and Endodontics, Guanghua School of Stomatology, Affiliated Stomatological Hospital, Sun Yat-sen University, Guangzhou, P.R. China, in order to verify a possible dental cause for a skin lesion. The patient’s chief complaint was the presence of a slightly stiff nodule in the right submandibular region, which had been periodically discharging pus for one year. She reported that the lesion had been previously diagnosed as a sebaceous cyst and had been surgically removed by a surgeon. Following its recurrence, for about seven months, she had undergone multiple failed regimens of traditional Chinese medical therapies. Finally, she was referred to a dermatologist and a dental aetiology was suspected. There was no history of dental pain, sensitivity or restricted mouth opening during the previous year. The patient’s medical history revealed no significant insights.Extra-oral examination revealed an extra-orally draining sinus and an erythematous, smooth, non-tender and slightly stiff nodule with crusting about 1 cm in diameter in the right submandibular region  and isolated with a rubber dam under local anesthesia. Working lengths were determined with an electronic apex locator, Raypex5 , and confirmed by diagnostic radiography Figure C. All caAt the next visit, about one month later, the extra-oral sinus tract had healed and no purulent discharge was noted Figure F. The caDental pathosis is the most common cause of cutaneous sinus tracts in the face and neck region and should be the primary suspect in differential diagnosis ,12. OtheOdontogenic cutaneous sinus tracts typically arise from periapical infections around the root apices as a result of pulpal necrosis, nearby caries or traumatic injury. Pulp sensitivity tests and radiographic analysis are the routine tests used to locate the involved teeth. Whereas the location of the sinus tract opening does not necessarily indicate the origin of the inflammatory exudate, tracking of the sinus tract with a gutta-percha point contributes to the final correct diagnosis ,13. HoweElimination of the infection is critical for the treatment of the draining sinus tract, either by endodontic therapy of restorable teeth or extraction of unrestorable teeth ,17. ExciThorough knowledge of both normal and abnormal root canal morphology contributes to successful endodontic treatment. The inability to locate, debride or fill all canals of the root canal system has been a major cause of post-treatment failure . MandibuSodium hypochlorite (NaOCl) is one of the most important and effective root canal irrigants due to its excellent bactericidal and tissue-dissolving properties. However, it also exhibits cytotoxicity and may cause severe tissue damage when extruded apically . NaOCl aDental infection should be considered a primary cause of cutaneous facial sinus tracts. In cases with restorable teeth, elimination of the infection through endodontic treatment leads to resolution of the sinus tract. Thorough knowledge of aberrant root canal anatomy is critical for infection management during root canal therapy. NaOCl accidents should be avoided when periapical bone destruction is significant, and CBCT imaging enables better evaluation of periapical bone destruction when evaluating the safety of NaOCl use. CBCT imaging facilitates successful endodontic treatment by aiding the diagnosis of odontogenic cutaneous sinus tract and enabling better understanding of unusual canal morphology.Written informed consent was obtained from the patient for publication of this Case report and any accompanying images. A copy of the written consent is available for review by the Editor-in Chief of this journal.


The dorsal nasal cyst formation is a rare and late complication of rhinoplasty. It has been rarely reported in the literature and it is usually mucous cysts. Migration and planting to the subcutaneous space during the surgical procedure has been recognized as the formation mechanism. This case report has presented 42-year-old male patient with a destructing dorsal nasal mucous cyst that developed 10 years after the rhinoplasty operation. There was no complication in the primary rhinoplasty and the patient was satisfied with his appearance. There was a swelling of the nasal dorsum over the past year and surgical excision of the cyst was performed. During the surgery, the defect was reconstructed with conchal cartilage. There was no recurrence during follow-up. The dorsal nasal cyst formation is a rare and late complication of rhinoplasty. Current literature contains only a few case reports about this complication and these are mostly mucous cysts .Different tissues such as bone, cartilage, and soft tissue can be affected during surgical procedures. Also, it is difficult to predict the outcome of surgery due to the dynamic nature of the healing process.The complications of rhinoplasty can be classified as early and late complications. Early complications are generally related to intraoperative sterility and surgical technique . Late coThe assumed mechanism of dorsal nasal cyst formation is thought to be the migration and seeding of surgical debridement to the subcutaneous tissue during surgery . In a feIn our study, we aimed to discuss a case about development of cystic formation in the nasal dorsum ten years after rhinoplasty.A forty-two-year-old man presented to us with right nasal radix mass which was increasing in size within the last year. The patient had undergone rhinoplasty surgery and there were no complications after the procedure. The physicial examination has revealed a 2 cm painless mass with smooth borders, cystic in nature, and it has caused erosion in the nasal dorsum through the nasal bone . Nasal tComputer tomography showed a heterogeneous mass which caused erosion in nasal dorsum through the nasal bone. Cranial MRI (T2A sequence) showed a cystic mass which was not spreaded into any intracranial structures Figures .The patient was informed about the risks and benefits of the procedure and informed consent was obtained from patient. After that, the operation was done under general anesthesia. Incision was made from sterile skin to the depth of the mass during operation. The mass, which eroded nasal dorsum and involed the nasal cavity, was dissected from surrounding tissues and entirely excised. A graft taken from conchal cartilage was laid over the defect according to its shape and then it was sutured to the surrounding tissue .The excised cystic tissue was well-circumscribed and contained brown, serous fluid. Final pathology result has reported that the tissue has an inflammatory polypoid formation with respiratory tract epithelium. No complications were observed during follow-up period. Also, no defects were observed cosmetically. The patient did not complain of any scar formation at his first year of check-up.Rhinoplasty is a very common surgery nowadays. The most important expectations of patients and surgeons from this surgery are pleasing from cosmetic results and no recurrence of any deformity over the years. However, many complications may develop even after years from rhinoplasty.Tissue edema, periorbital ecchymosis, infection, skin necrosis, and numbness can be seen in the early period after rhinoplasty surgery. Early period deformities can occur mostly due to mistakes in surgical technique. The most important cause of late period revison surgery is postoperative deformities which composes of approximately 5–15% of all causes .The development of mucous cyst formation is a rare complication in the late period. Generally, it can be observed in the nasal dorsum along the nasal osteotomy line. The migration and seeding into the subcutaneous space was accepted by authors as the mechanism of injury. It has been believed that this usually occurs after a traumatic surgical technique. Karapantzos et al.  have repAll cyst cases have supported the seeding theory till now and then they were characterized as being successfully treated with surgery without any recurrence.Interestingly, one case report has reported a cyst that developed from mandibular respiratory epithelium in patient underwent genioplasty and rhinoplasty surgery. In this case, it has shown that using osteocartilaginous material for chin augmentation caused the implantation .The most important way to avoid these complications is preparing the graft by separating it from the epithelium as much as possible and avoiding unnecessary trauma during the osteotomy.After rhinoplasty, developed cysts are usually mucoid in nature and arise from respiratory epithelium. However, a case has been reported about development of epidermoid inclusion cysts in posttraumatic patients after rhinoplasty.The most important point in the treatment of developed dorsal nasal cyst is complete excision of cyst and reconstruction of nasal dorsum . In thisThe most important issue for used graft materials on the nasal dorsum is causing the irregularities that can be seen from the outside. Therefore, graft material should be carefully selected and placed properly.Septal, costal, or conchal cartilage can be used for reconstruction. In this case, our patient previously had septoplasty; thus he did not have sufficient cartilage remaining; therefore, we used the conchal cartilage for reconstruction surgery. Nasal dorsum was supported with autogen chonchal graft. By this way, we have created a natural cosmetic appearance. The conchal cartilage was slightly sloped and erosional area was also sloped. So it was made in a better way.The dorsal nasal cyst formation is a rare and late complication of rhinoplasty. The most important thing to avoid this complication is to be sure from cleaning all of the epithelium from the prepared graft and avoiding any unnecessary trauma. Complications seen after rhinoplasty can be reduced by increased surgical experience.