We report the first case of spontaneous infarction occurring in pleomorphic adenoma located in infratemporal fossa mimicking parapharyngeal abscess, including clinical presentation, workups, and surgical approach. The final diagnosis was confirmed on a histological basis. The combination of CT scan and MRI might be helpful in distinguishing neoplasia from abscess collection in this area. Surgical intervention is the treatment of choice. PA is the most common salivary gland neoplasia that also can occur in the respiratory tract or nasal cavity .Although infarction of PA after fine-needle aspiration has been documented thoroughly, unprovoked infarction of PA has remained as an unusual entity reported in the literature . To our The infratemporal fossa (ITF) is located posterolaterally to the maxilla and maxillary antrum. It is bounded anteriorly by the maxilla and posteriorly by the glenoid fossa as well as the mandible. Medially, the ITF is bounded by the lateral pterygoid plates. The roof contains the foramen ovale and foramen spinosum and contains the pterygoid muscles.Neoplasia involving the ITF region may arise from tissues in the region. However, more often, they result from the extension of the surrounding structures. Rarely, metastatic lesions can be seen in this region. Due to its concealed localization, tumors may remain unnoticed for quite some time; thus, signs and symptoms often appear late, insidious and may be falsely attributed to other disease processes .2A 20-year-old Mediterranean housewife female patient presented walk into the emergency department with a 5-day history of severe sore throat associated with difficulty of swallowing and trismus, which was not relieved with supportive treatments and oral antibiotics. Apart from minor thalassemia, her medical history was insignificant, and insignificant past surgical history; patient denied having fever, weight loss, night sweat, and loss of appetite, patient has no family history of malignancy, anyhow she reports multiple similar episodes of recurrent sore throat that responded to co-amoxiclav oral antibiotic.Clinical examination revealed a left-sided peritonsillar swelling with a mild deviation of the uvula to the right side; she also had painful and tender submandibular lymph nodes on the left side as well. The fiberoptic exam showed mild medialization of the left parapharyngeal wall. Major salivary glands examination was unremarkable. Her routine blood tests were within normal limits except for the picture of microcytic hypovolemic anemia consistent with her thalassemia condition and elevated C-reactive protein (CRP) that was 28.9 (WBC count6.0 with no leukocytosis). Suspected to have a peritonsillar collection; aspiration was attempted in the emergency room that, however, yielded no pus. A computerized tomography (CT) neck with contrast  was doneThe patient shifted to the operating theater for incision and drainage of left parapharyngeal abscess; trans-cervical approach was made initially by the ORL-HNS consultant to drain the suspected abscess. However, no pus yielded, repeated aspiration also did not yield anything. Therefore, a trans-oral approach followed where incision anterior to the anterior tonsillar pillar was done. With further dissection until reaching the left infratemporal fossa, the upper pole of a mass was noticed and was hard, surrounded by necrotic tissue. This full mass was delivered completely through transoral incision.Post-operative, the patient tolerated the surgery very well and started diet short after the operation without any limitations; she was discharged on postoperative day 3 with analgesia.The patient followed for 1 year and she was doing fine without any signs of recurrence, neurological deficit or palatal dysfunctional issues; no further imaging workup was done for the patient as the progression of the disease along with the final pathological report indicate a benign pathology.Histopathological; grossly, the specimen consisted of a single irregular firm tan brown soft tissue nodule measuring 4 × 3 × 2.5 cm. Cut surfaces showed brown heterogeneous soft tissue with multiple scattered white firm parts largest measured 1.5 × 1.5 × 0.5 cm abutting surgical margin .Fig. 2ReMicroscopically, the extensively sampled specimen showed myoepithelial cells scattered within myxoid stroma intermingled with sheets of degenerate ductal epithelial cells together with prominent squamous metaplasia and abundant keratinization.Focal hemorrhage, inflammation, and necrosis were also noted. A rim of normal salivary gland tissue was identified. The overall appearances were consistent with an infarcted pleomorphic adenoma with florid squamous metaplasia . No mali3Tumors of the ITF present a diagnostic and a surgical challenge due to the often-occult nature of such lesions as well as the complexity of the local anatomy. Neoplasia of ITF can be classified as primary, secondary or metastatic . Among tCT scan remains a substantial image modality in diagnosing tumors of the ITF as it helps to determine the extent of the disease, its local spread, and radiologic characteristics of the tumor may help to some extent in determining the type of the tumor.Furthermore, Magnetic resonance imaging (MRI) should be considered as a modality whenever the suspicion of the vascular lesion is present or in cases of atypical presentation as in our case, the palate is the most common site of minor salivary gland PA followed by the lip, buccal mucosa and floor of the mouth, tongue, tonsil pharynx, retromandibular area and nasal cavity, parapharyngeal space PA can originate de novo or from the deep lobe of the parotid gland and then extend by the stylomandibular tunnel into the parapharyngeal space.Parapharyngeal tumors are rare, constituting less than 0.5% of head and neck neoplasms . Of thesDue to its concealed location, lesions often appear late; in addition, surgical planning is confounded by proximity to vital structures, namely intracranial structures, the orbit, sinuses, and the nasopharynx. Indicating the difficulty of access, numerous surgical approaches have been described.Although the prognosis of PA is generally good, yet the conventional watchful waiting and clinical follow up is not sufficient, while periodic radiological evaluation might be needed to detect any recurrence post-surgical excision.While many surgeons acclaim complete surgical excision is the only valid treatment option for pleomorphic adenoma, other reports illustrated good results with adjuvant radiotherapy against incompletely resected tumors due to its location that renders them inoperable, However, this remains a matter of controversy .Although seems to be relatively rare, spontaneous infarction resembling abscess formation should be taken into consideration in the differential diagnosis of such cases. In atypical case presentations, one might consider MRI to differentiate between abscess and neoplasia in the ITF region. This case has been reported in line with the SCARE Guideline 2018 .4ITF Infarcted neoplasms may present clinically as an abscess, in these situations, the combination of CT scan and MRI might be a helpful diagnostic tool in distinguishing neoplasia from abscess collection in this area. Treatment of ITF tumors is surgical. The transcervical approach allows excellent control of the tumor and neurovascular elements but the transoral approach might be also needed according to the case nature.No conflict of interest.Non sponsored and no fund received.The paper describes a case report therefore no special ethical consideration required.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.ARA: Data Collection, Literature Search, Manuscript Preparation,AAA: Manuscript Preparation & revision,HAH, WR: Manuscript Preparation,BAW: pathology slides preparation,AJN: Manuscript Revision and submission.All authors read the final manuscript and agreed on it.N/A.Dr Abdelrahman R. AlsalehAAlsaleh@hamad.qa00974-55176528Not commissioned, externally peer-reviewed.


Invasive cervical resorption (ICR) is an uncommon phenomenon (0.1%), however, it represents a challenge to the structural and functional integrity of the dentogingival junction, as well as a risk for the survival of the affected tooth. They are characterized by their location and invasive character, being able to appear in any tooth of the permanent dentition. It shows up after the damage to the cervical insertion apparatus, leaving the pulp without participation in the origin of the lesion. They may appear just below the junctional epithelium or at a more apical level. The MTA®  and the Biodentine®  are two biomaterials that have demonstrated the ability to promote the neoformation of cement so they are considered an alternative in the treatment of the ICR. This article presents an ICR clinical cases treated with these biomaterials, in which favorable post-operative healing is observed. Key words:MTA, Biodentine, biomaterials, root resorption, invasive cervical resorption. Root resorption (RR) is defined as the loss of tooth hard tissue (dentin and cement), through the action of osteoclasts . These cThe pathway RANK (associated receptor with nuclear factor Kappa Beta) / RANK-L (the respective ligand) / OPG (Osteoprotegerin), is related to the osteoclasts activation. RANK-L, attached to another ligand protein (M-CSF), promotes the formation and activation of clastic cells which leads to increased bone resorption. This process, closely related to bone remodeling processes, is very similar to the ones that occurred in the RR. The periodontal ligament (PL) cells, pulp cells, odontoclasts and cementoclasts present OPG, which inhibits the clastic action of RANK-L . WithoutWhen speaking of RR in a pathological context, there must be a stimulus, which origin could be pulpal or bacterial. This could trigger a benign fibrovascular proliferation or a fibro-osseous disorder . The absLinskog (2006) makes a classification of root resorptions, dividing them into 3 main types: by trauma, infection or hyperplastic, with the ICR in the last one.In this paper we focus on cervical invasive resorption, so we will briefly review the characteristics. Hyperplastic reabsorption - the group to which the ICR belongs  - has anICR is a rare form of external RR with a prevalence of 0.1% . They arIn a clinical view, it is sometimes characterized by the presence of a “pink tooth” or “Mummery tooth“  as the highly invasive tissue becomes visible because of its high vascularity. If there is no external clinical signs, the diagnosis will be made by X-rays. Patients usually do not refer discomfort unless a secondary infections with pulpal or periodontal symptoms are present. The cause of this condition is uncertain, although predisposing factors such as trauma, internal bleaching or orthodontic treatment have been identified. These can alter or damage the cement bringing on the appearance of the process.The treatment can vary depending on the progression degree of the disease, although for a correct treatment the entire inflammatory tissue must be removed and the restoration of the defect must be achieved in order to allow the attachment of the dento-gingival complex. ICRs are divided into 4 categories  dependinPrecisely its location causes a situation of risk for the bacterial and immunological homeostasis of the oral cavity, since there is a destabilization in the nexus between the oral and internal environment. Although the active defense capacity of the epithelium and its cells is known, by the synthesis of defensins, calprotectin and lysosomes, the absence of a physical seal by the epithelium of attachment to the tooth supposes a situation of periodontal risk . As indiin vitro studies , with a curing period of up to 21 days, compromising its handling difficulty. Another material used in the treatment of reabsorbed processes is the Biodentine®. This is a calcium silicate cement, with a much shorter setting time (12 minutes) being the main advantage respect to the MTA®. One study observed that fibroblasts proliferate in the presence of Biodentine®  and thatFollowing is a series of cases with ICR treated with these materials and their subsequent evolution.Case 1: 50-year-old patient with no medical history of interest who, after radiographic and clinical examination, was detected an external resorption in the coronal third of the root of tooth 3.5, classified as grade II . InitialCase 2: 61-year-old patient with no medical history of interest who was diagnosed with ICR on tooth 2.2. An sulcular incision was made, exposing and debriding the tooth and the root, and then the MTA® was placed. In this case, due to the extension of the lesion that was determined as Grade II (Case 3: 62-year-old patient, without medical history of interest. ICR was diagnosed in the palatine of tooth 1.2. A palatine approach was made with a mucoperiosteal flap for the exposure of the lesion and its curettage. It was filled with Biodentine® and sutured. Endodontic treatment was performed due to the degree of involvement of the piece, Grade II (Case 4: 14-year-old patient, with no medical history of interest, who was diagnosed with an ICR on tooth 1.1, with previous endodontic treatment due to post-traumatic necrosis. An unfavorable evolution was observed at the age of 18. At that time, it was diagnosed as Grade III . IntrasuThis paper presents the evolution of 4 cases that present ICR, with grades II and III . After tLonger observation and evaluation periods are needed, in order to assess the long-term stability of these solutions. Nevertheless, the high biocompatibility of this material seems to offer good perspectives. It would be interesting to deepen the investigation of the periodontal tissues response, more specifically the dentogingival junction in contact with these biomaterials.


Perioperative chemotherapy could improve oncological outcomes for patients with advanced colon cancer. However, the effectiveness of triplet chemotherapy in the neoadjuvant setting is still unknown.A 61-year-old man was referred to our hospital due to abdominal distention. Abdominal computed tomography showed a huge, 18-cm mass in the right upper abdomen. Biopsy showed well-differentiated adenocarcinoma. Locally advanced transverse colon cancer T4b N2a M0 Stage IIIC was diagnosed. Considering the extensive invasion to surrounding organs and difficulties in achieving margin-negative surgery, an emergency ileostomy was performed first. Then, neoadjuvant chemotherapy (NAC) consisting of a combination of 5-fluorouracil (5-FU), oxaliplatin, irinotecan, and leucovorin (FOLFOXIRI) was planned, followed by primary tumor resection. After 6 courses of treatment, the primary tumor shrank remarkably. Finally, laparoscopic radical extended right hemi-colectomy was performed. There were no residual tumor cells in resected specimens, including the primary tumor and surrounding lymph nodes. The pathological diagnosis was complete response.A case of pathological complete response after neoadjuvant treatment followed by radical resection was reported. Further research is needed to confirm the appropriate indications for neoadjuvant FOLFOXIRI therapy for patients with LACC. HoweverRecently, laparoscopic surgery has become the standard approach for colon cancer, with better short-term outcomes including less blood loss, quicker recovery of bowel function, and less postoperative complications . HoweverNeoadjuvant treatment is expected to have advantages such as tumor regression, tumor shrinkage, and down staging, making successful laparoscopic surgery more likely . FurtherA case of pathological complete response (pCR) of locally advanced transverse colon cancer after preoperative FOLFIXIRI therapy followed by laparoscopic right hemi-colectomy is reported.This work is reported in line with the SCARE criteria .2A 61-year-old man was referred to our hospital due to abdominal distention. Physical examination demonstrated a huge, hard, tender mass in the right upper quadrant of the abdomen. Laboratory data showed inflammation , anemia , malnutrition , and an elevated tumor marker (carcinoembryonic antigen 64.1 U/mL). Abdominal computed tomography showed a huge 18-cm mass in the right upper abdomen . The tum3In the present case report, a pCR was achieved after neoadjuvant FOLFOXIRI plus bevacizumab for LACC.Several guidelines recommend wide surgical resection as the treatment strategy for LACC ,12. FollSome RCTs reported that laparoscopic surgery had less blood loss, shorter time to pass first flatus, less use of analgesics after surgery, and a shorter hospital stay ,17. For the FOxTROT trial, which aimed to investigate the feasibility, safety, and efficacy of NAC for colon cancer, was reported [The major potential drawback of NAC is that disease progression during NAC may preclude curative surgery. In regard to LACC patients, several studies have evaluated the effect of NAC, and they reported that NAC is safe and feasible ,9,18,19.In the FOxTROT trial, the patients underwent 3 courses (six weeks) of NAC followed by surgery . In the However, the appropriate timing of surgery during triplet chemotherapy is not well known. A further large-scale study is needed to clarify this issue.There is no consensus about which regimen, doublet or triplet, should be selected for NAC. Several clinical trials selected doublet therapy using oxaliplatin and fluoropyrimidine as NAC for LACC patients, and overall response rates ranged from 13.6% to 30.8%, and pCR ranged from 0 % to 6.8 % ,9,18,19.On the other hand, severe adverse events including neutropenia, diarrhea, and nausea occurred significantly more often in patients receiving triplet therapy compared to doublet therapy ,23. In tThe best method of follow-up for colon cancer patients with pCR after neoadjuvant treatment is controversial. In rectal cancer, distant metastasis still remains a concern in pCR patients after chemoradiotherapy . Therefo4A case of pCR after neoadjuvant treatment followed by radical resection was reported. Further research is needed to confirm the appropriate indications for neoadjuvant FOLFOXIRI therapy for patients with LACC.NoneNoneEthical approval has been exempted by our institutionWritten informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on requestTetsuro Tominaga and Takashi Nonaka conceptualized the study. Kazuhiro Tabata made a pathological diagnosis.Terumitsu Sawai and Takeshi Nagayasu provided input on the manuscriptResearchregistry1025Tetsuro TominagaProvenance and peer reviewNot commissioned, externally peer-reviewed


Giant hydronephrosis (GH) is a rare disease that is found in adult patients. Although there are some common symptoms associated with hydronephrosis, such as surrounding organ compressed, its rarer symptoms can render diagnosis very difficult, and treatment should also vary according to the cause.We here report an 82-year-old man who was admitted to the hospital for repeated intractable hiccups. After B-ultrasound and CT examination, the patient underwent laparoscopy surgery, which was converted to open nephrectomy, and the patient’s intractable hiccup symptoms disappeared.GH is a rare disease, and its symptoms are diverse. The more unusual symptoms of cystic hypertonic compression of surrounding organs, such as intractable hiccups, should be taken into account. GH is mainly diagnosed via ultrasound examination and CT scan. The choice of treatment for GH needs to be based on the etiology and renal function of hydronephrosis, and consider malignant lesions.Giant hydronephrosis can present rare symptoms as “intractable hiccups”. The selection of treatment should be made depending on the cause. In adults, GH is defined by Sterling firstly in 1939 as the presence of more than 1 L of fluid in the renal pelvis, or kidney occupying the hemiabdomen across the midline [GH may present with vague symptoms, including increased abdominal girth, nausea, fatigue, indigestion, and loss of weight, but, to the best of our knowledge, there have been no reports of intractable hiccups due to GH. We here present a rare case of an 82-year-old male patient who suffered from intractable hiccups due to GH, along with a review of the past decade. This case has been reported in accordance with the surgical case report guidelines (SCARE) criteria [2An 82-year-old man complained of a gradual increase in his abdominal girth over the past two years and of abdominal distension and intermittent nausea for the past six months. He was admitted to the hospital for repeated intractable hiccups having lasted two months. The patient had a history of intestinal necrosis due to an intestinal obstruction 25 years earlier and of occlusion of the inferior vena cava filter and left common iliac vein stent placement due to left common venous thrombosis five years earlier. Physical examination showed the patient was of average build with a thin, anemic appearance, and his vital signs were stable. Cardiopulmonary examination produced normal results. An abdominal examination revealed a longitudinal surgical scar in the middle of the upper abdomen. The swelling of the mass was evident on the left side of the abdomen . PalpatiAfter administration of two units of homologous red blood cells, the patient underwent what was initially laparoscopy and then open simple nephrectomy. Adhesion between the kidney and surrounding organs was evident during the procedure. About 7.5 liters of hydronephrosis fluid were drained. The left kidney and upper ureter were successfully removed . The pat3GH is a relatively rare urinary system disorder that can occur in patients of any age, but it is extraordinarily rare in adult patients . It is gGH progresses slowly. As described in the literature, the associated massive abdominal mass or abdominal swelling may cause pain, hematuria, recurrent urinary tract infections, or other symptoms or complications described in the literature, including nausea, fatigue or indigestion, urinary tract infection, weight loss, renal insufficiency, and even kidney breakdown . AccordiAs far as we know, no previous reports have shown any case of hiccups caused by massive hydronephrosis. Hiccups are an abnormal respiratory movement, a diaphragmatic spasm that occurs mainly because of vagus nerve reflex or direct stimulation of the phrenic nerve and diaphragm, which causes the diaphragm and intercostal muscle to involuntarily and synchronously contract. This instantly produces strong inspiratory movement. Intractable hiccups is defined if the attack lasts more than 1 month, and there are various factors that can cause intractable hiccups, including metabolic abnormalities, psychogenic diseases, malignant tumors, central nervous system pathology, medications, pulmonary disease, and gastrointestinal conditions . In thisGH is mainly diagnosed via ultrasound examination and CT scan ,13. In mThe treatment of GH involves nephrectomy if the kidneys are nonfunctional; for functional organs, the treatment includes percutaneous nephrostomy, pyeloplasty, renal cortical fold repair, and occasionally nephrectomy . PatientIn conclusion, GH is a rare disease, and its symptoms are diverse. The rarer symptoms of cystic hypertonic compression of surrounding organs, such as intractable hiccups, should be taken into account. Treatment of GH includes percutaneous nephrostomy, pyeloplasty, renal cortical fold repair for functional kidneys, and nephrotomy for non-functional kidneys. This case and literature review showed that GH combined with malignant tumors of the renal pelvis is common, so unless it can be diagnosed as benign GH, malignant lesions should be taken into account when planning treatment, so as to prevent postoperative tumor recurrence and metastasis .Beijing Council of Science and Technology “Special Project for Applied Research of Capital Clinical Features” (NO Z171100001017131).The authors declared that this study supported by All authors have issued final approvals for the version to be submitted.This study is exempt form ethical approval.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.Xiaoxing Liao performed the surgical procedure, proposed the study and wrote the paper.Jianhua Yang collected data and pictures from surgery.Nianzeng Xing supervised the paper and controlled all the analysis of results, including language.Not needed.Dr.Xiaoxing Xiao.Dr.Nianzeng Xing.Not commissioned, externally peer-reviewed.No conflicts of interest.


Adrenocortical carcinoma (ACC) is a rare endocrine malignancy, often with an unfavorable prognosis. Radical adrenalectomy is the gold standard of treatment of localized disease.We report a case of a 23-year-old male patient who presented with persistent left flank pain and urticaria for 3 months. Imaging studies confirmed the presence of a large left adrenal mass with malignant features. The biochemical workup was unremarkable. Open left radical adrenalectomy was performed, the final pathologic examination showed ACC with negative surgical margins. The patient remained disease-free for eighteen months period of follow up after surgery.ACC is a rare neoplasm with poor prognosis and with an incidence of one in one million population. There is a slight female predilection. The ACC may be functional with a clinically pure endocrine syndrome like Cushing syndrome. Most of patients with ACC present with symptoms and signs of hormonal secretion. Adrenal computed tomography (CT) scanning and magnetic resonance imaging (MRI) are the imaging studies of choice in ACC. When feasible, total resection remains the treatment of choice for the definitive treatment of ACC. The benefit of the use of mitotane as an adjuvant treatment has been considered controversial. Adjuvant mitotane significantly decreases the recurrence and mortality rate after resection of ACC in patients without distant metastasis as proved by some studies, but these findings need further validation.ACC is a rare neoplasm characterized by a high risk of recurrence after surgical resection. Most ACCs cause hypersecretion of steroids including glucocorticoids and androgens. ACC patients have a very poor prognosis with a 5-year overall survival below 30% in most series [Patients usually present with hormonal hypersecretion signs  or mass effect. Tumors typically appear inhomogeneous in both computerized tomography and magnetic resonance imaging with necrosis and irregular borders and differ from benign adenomas by their low fat content .In patients with suspected localized ACC, a thorough endocrine and imaging workup is followed by complete resection of the tumor by an expert surgeon. In patients with advanced disease at presentation or recurrence not amenable to complete resection, mitotane alone or in combination with cytotoxic drugs is the treatment of choice .This work has been reported in accordance with the SCARE criteria .2A 23 years old male patient, Know to have poorly controlled diabetes mellitus type 1 presented to the outpatient clinic with 3 months history of left flank pain. His pain was associated with a weight loss of 8 kg. The patient denied any recent sexually transmitted disease or genitourinary trauma. He is a non-smoker and non-alcoholic.On presentation, his temperature was 37 °C and the vital signs were within normal range. Physical examination revealed, severe left Costovertebral angle tenderness. Skin observation revealed blancheable, erythematous, oedematous papules or ‘weals’ identified on the back and buttock . His urtLaboratory examination revealed WBC of 5000/mm3 with normal eosinophils, CRP of 2 mg/L, random blood sugar 200 mg/dl. Urine analysis showed 1–2 WBC per high power field. Serum creatinine was 1.7 mg/dL. Skin patch test was negative.Computed tomography (CT) scan of the abdomen and pelvis without IV contrast showed an 8 × 5 cm mass in the left adrenal gland . MRI of Biochemical workup  was unremarkable aside from plasma dehydroepiandrosterone-sulphate (DHEA-S) level was 120.6 mg/dL . A metastatic workup including a Computed tomography (CT) of the chest and a bone scan was negative for metastases.Surgical resection was recommended and the patient underwent open left adrenalectomy. There was no evidence of gross invasion of the surrounding structure intraoperatively.Surgical pathology confirmed the diagnosis of adrenocortical carcinoma, stage II . SurgicaThe patient was discharged 3 days post surgery. He was seen in the outpatient clinic 4 weeks after discharge and he had marked improvement with the resolution of his urticaria. The clinical and radiologic follow-up with thoraco-abdominopelvic CT for 6, 12 and 18months showed no signs of local recurrence and distant metastases.The patient provided a written consent for the publication of this clinical case.3ACC, a rare and aggressive malignancy, accounts for up to 14% of adrenal incidentalomas. It has a bimodal age distribution with a peak in the first decade and a peak in adulthood in the fourth and fifth decades. Women are most frequently affected. The most important predictors of survival in these patients are tumor grade, tumor stage, and surgical resection .Several genes have been implicated as tumor drivers in sporadic ACC, including mutations in insulin-like growth factor 2 (IGF2), β-catenin (CTNNB1 or ZNRF3), and TP53. Other germline variants of some of the same genes identified to be drivers of sporadic ACC are also associated with familial tumor syndromes characterized by ACC, including Beckwith-Wiedemann syndrome (BWS), familial adenomatous polyposis (FAP), and Li-Fraumeni syndrome .Most patients (40–60%) present with steroid hormone excess  or abdominal mass effects (30%), but 15–20% of patients with ACC are initially diagnosed incidentally .Contrast-enhanced CT or MRI is the diagnostic imaging modality of choice for initial imaging and staging as well as for follow-up. The typical appearance of ACC on unenhanced CT is a large, inhomogeneous but well-defined suprarenal mass that displaces adjacent structures as it grows. Regions of low attenuation correspond pathologically to necrosis. ACC is typically heterogeneous in signal intensity on MRI because of the presence of hemorrhage and/or necrosis on T1-weighted imaging, ACC is typically isointense or slightly hypointense to normal liver parenchyma, whereas, on T2-weighted imaging, ACC is usually hyperintense to liver parenchyma .The European Network for the Study of Adrenal Tumors (ENSAT) classification, has been proposed. In this system, stage I and stage II as strictly localized tumors with a size of ≤5 or >5 cm. Stage III ACC is defined by the presence of positive lymph nodes, infiltration of surrounding tissue, or tumor thrombus in the vena cava and/or renal vein; and stage IV ACC is restricted to patients with distant metastasis .Currently, the only curative approach to ACC is complete tumor resection. Adjuvant therapies aim to decrease the chance of recurrence . LaparosACC is a rare neoplasm characterized by a high risk of recurrence after radical resection. Whether the use of mitotane is beneficial or not as an adjuvant treatment has been controversial. A large retrospective study conducted by Terzolo M et al., suggests a benefit associated with the use of adjuvant mitotane therapy after radical resection of adrenocortical carcinoma . ConsideAdrenolytic therapy with mitotane administered alone or in combination with other antineoplastic agents is the primary treatment for patients with advanced ACC . RecentlInsulin-like growth factor 1 receptor signaling through upregulation of the stimulatory ligand IGF-II has been implicated in the pathogenesis of ACC. A study investigated figtilimumab, a fully human monoclonal antibody directed toward IGF-1R in patients with refractory adrenocortical carcinoma. Eight of 14 patients had a stable disease as their best response . AlthougWe present a rare case of ACC in a young patient with nonfunctioning tumor. We also proved that that complete surgical resection (R0 excision) offers the best chance for long-term survival in patients with stage II ACC. In our case, there was no evidence of recurrent or metastatic lesions during the follow-up period of 18 months. We also demonstrated the possible association of the malignant disease with chronic urticaria. Dehydroepiandrosterone and its sulfate derivative (DHEA-S) appear to have regulatory effects on immune homeostasis.Patient’s urticaria may be related to the decreased serum concentration of DHEA-S level. There are several hypothetical explanations of the phenomenon of lower circulating concentration of DHEA-S in chronic urticaria patients. It has been suggested that during chronic inflammatory response and severe stress conditions, a hormonal shift may occur in the adrenal steroid production into the direction of cortisol relative to DHEA-S, which is probably necessary to achieve adequate cortisol level at the expense of adrenal androgens. However, current knowledge prevents answering whether lower circulating DHEA-S concentration is a primary phenomenon or just an accompanying one which appears as a response of different systems to the course of the illness .4ACC is an uncommon malignancy with high recurrence and mortality rates. ACC is often diagnosed at an advanced stage. A multidisciplinary team approach and discussion can provide the best management and prognosis for ACC diagnosed patients.No fundingEthical approval is not required by our institution.Written informed consent was obtained from the patient for publication of this case report and accompanying images.Yasmin Moussa, Mohamed Abou Chakra, Mohamad Moussa: Case report design.Yasmin Moussa, Mohamed Abou Chakra, Mohamad Moussa: Manuscript preparation.Mohamed Abou Chakra, Mohamad Moussa: Followed up the patient and revised the manuscript.Yasmin Moussa, Mohamed Abou Chakra, Mohamad Moussa: Approved the final manuscript.Not applicable, case report.Mohamed Abou chakra.Not commissioned, externally peer-reviewed.None.


Regarding the radiology of our two cases, the humeral lesion appeared almost entirely intramedullary without soft tissue extension; the midfoot lesion demonstrated a destructive, metatarsal-centered bone lesion, initially thought clinically to represent primary bone osteosarcoma. The diagnoses of monophasic synovial sarcoma were rendered via core needle biopsies, with molecular FISH confirmation of SYT gene rearrangement. Clinical follow-up data was only available for the female patient with the primary humeral lesion, who underwent surgical resection, with no local recurrence or distant metastasis at 7 months postsurgery. To our knowledge, these are the first reported examples of molecularly confirmed, primary intraosseous synovial sarcomas of the humerus and metatarsal bones. Primary intraosseous synovial sarcomas with molecular confirmation differ clinically from those lacking it; however, the demographic features and metastatic potential appear similar to primary soft tissue synovial sarcoma.Synovial sarcoma is a well-known malignant tumor usually originating within deep soft tissues of the lower extremities of adolescents and young adults. Rare radiologically confirmed examples of primary bone synovial sarcoma have been documented, generally in isolated case reports. Herein, we report two cases of primary intraosseous synovial sarcoma, with molecular confirmation, involving the left humerus of a 45-year-old female and the right fourth metatarsal bone in a 36-year-old male. Additionally, we clarify the spectrum of primary intraosseous synovial sarcoma by separately analyzing reported cases with radiographic confirmation of bone origin and molecular support for the diagnosis. There are clinicopathologic differences between those tumors with documented molecular confirmation and those lacking such confirmation, specifically regarding their anatomic distribution ( SYT gene on chromosome 18 and either the SSX1 or SSX2 gene on chromosome X, is identified in the overwhelming majority (over 90%) of synovial sarcomas [Synovial sarcoma is a malignant mesenchymal neoplasm most commonly arising in the deep soft tissues of the extremities in young to middle-aged adults. It accounts for approximately 5-10% of soft tissue sarcomas, often arising near joint spaces, and very rarely presents primary to the viscera or bone [sarcomas , 7, radiity over 0% of synsarcomas . An addisarcomas –22. AmonThis study was approved by the Cleveland Clinic Institutional Review Board (IRB#19-680). The in-house and consultative files of 2 coauthors (J.R. and S.K.) were reviewed for diagnoses of primary intraosseous synovial sarcoma. Routine hematoxylin and eosin stained slides were rereviewed to confirm diagnoses. For inclusions, tumors had to demonstrate a clearly unequivocal intraosseous epicenter and had to have undergone molecular confirmation of the rendered histologic diagnosis. Clinical and radiographic features were also rereviewed and compared. Treatment and clinical follow-up data were obtained from the patient files, when possible. Extensive review of the English language literature within the PubMed database, without restriction based on date of publication, was performed to identify previously reported cases of intraosseous synovial sarcoma with and without molecular confirmation of the diagnosis. Fisher's exact test was performed to assess for statistical significance between molecularly confirmed and non-molecularly confirmed cases of intraosseous synovial sarcoma regarding anatomic site distribution. Statistical analysis was performed using online statistical software .Two cases were identified. Case presentation details are below.A 45-year-old female presented with a 2-year history of intermittent and worsening left shoulder pain, which recently began radiating to her left fingers and was associated with numbness and restricted range of motion. She reported physical aggravation to the area 3 days prior to presentation, secondary to moving luggage. There was no prior history of malignancy. Physical examination demonstrated limited active range of motion secondary to pain as well as tenderness to palpation over the left bicipital groove. No external abnormalities or palpable masses were noted. Magnetic resonance imaging (MRI) revealed a marrow-replacing lesion involving the proximal 6 cm of her left humerus , with associated periostitis, but minimal cortical destruction and no definite soft tissue extension . It was SYT (SS18) gene rearrangement, performed at the Cleveland Clinic on biopsy material that did not undergo decalcification, was positively confirmed  gene rearrangement, performed at the Cleveland Clinic, was positively confirmed . Reported cases with molecular/cytogenetic confirmation had an average patient age of 43 years  and a male : female ratio of 2.25, while those cases lacking molecular/cytogenetic confirmation had an average patient age of 34.8 years  and a male : female ratio of 0.6. Those with molecularly/cytogenetically confirmed primary intraosseous synovial sarcoma had the disease nearly exclusively limited to the long bones of the appendicular skeleton (excluding 1 case involving a thoracic vertebra), most commonly the lower extremities, with the tibia representing the single most common anatomic site. Cases lacking molecular/cytogenetic confirmation predominantly involved the head and neck, specifically the mandible and maxilla . To our knowledge, not a single case of primary intraosseous synovial sarcoma of the head and neck has ever been confirmed via molecular or cytogenetic means.When comparing the demographic data of molecularly/cytogenetically confirmed primary intraosseous synovial sarcomas with reported cases lacking this confirmation, it is interesting to note some differences in mean patient age, sex ratios, and anatomic distributions  had relevant reported clinical management data regarding their initial presentation, and 9 (69.2%) had reported clinical follow-up data. Of 11 patients, 6 (54.5%) reportedly received adjuvant or neoadjuvant chemotherapy .6% had r, 11. In At least some examples of what was originally thought to represent primary bone hemangiopericytoma  now appear to be misdiagnosed synovial sarcomas . Both ofp < 0.0001) in anatomic site distribution between those with and those lacking molecular confirmation of diagnosis. Given the rarity of the disease, establishing an accurate diagnosis of intraosseous synovial sarcoma necessitates radiographic correlation coupled with judicious use of ancillary studies especially molecular testing, such as FISH analysis, to ensure an accurate diagnosis.In summary, primary intraosseous synovial sarcoma, with supportive radiologic and molecular and/or cytogenetic confirmation, is an extraordinarily rare entity, most commonly arising in the appendicular skeleton, especially the long bones of the lower extremities . The vast majority are lytic, destructive lesions, usually associated with soft tissue extension. Histologically, most are monophasic spindled cell (fibrous). To our knowledge, including our cases, there are only 13 molecularly confirmed examples in the literature. We have described the first cases, also with molecular confirmation, of primary humeral and primary metatarsal intraosseous synovial sarcoma. It appears that cases of primary intraosseous synovial sarcoma with molecular or cytogenetic confirmation of the diagnosis, though limited in number, have demographic features and metastatic potential similar to primary soft tissue synovial sarcoma, demonstrating a male predominance and a rate of metastasis nearing 40%. Regarding overall prognosis, the paucity of bona fide cases combined with the limited available clinical follow-up data precludes definitive conclusions. Reported examples of intraosseous synovial sarcoma without molecular confirmation may represent a diverse group of neoplasms, many of which may not be “true” synovial sarcoma, which is highlighted by the significant difference (


Isolated primary sacral diffuse large B cell non-Hodgkin’s lymphoma is a very rare entity, and only 11 cases have been reported previously.A 36-year-old man was referred with low backache and radiculopathy pain with a clinico-radiological and cytological diagnosis of sacral metastasis. Histopathological examination and immunohistochemistry of image-guided tissue core biopsy from the sacral mass confirmed it as high-grade diffuse large B cell lymphoma (DLBCL). With normal blood counts and bone marrow, and no lesions elsewhere on imaging, he was staged IAE and received 6 cycles of R-CHOP  regimen chemotherapy followed by radiotherapy. The patient has completed a 3-year follow-up and is doing well with yearly imaging showing no evidence of active disease or recurrence.The case shows the importance of an image-guided core biopsy and immunohistochemistry over a fine needle aspiration cytology in select cases as it can alter the treatment and outcome in patients. Because of rarity, the treatment and prognosis in primary sacral NHL is not still very clear as it is treated as per the guidelines of treatment of bone lymphoma. Primary bone lymphoma is a rare entity constituting less than 2% of all lymphomas in the adult population; it usually involves long bones of extremities , 2. The A 36-year-old man was referred to us with a 30-day history of imbalance while walking, lower backache, tingling, and numbness in the left gluteal region radiating to the left lower limb. He was evaluated outside with a contrast-enhanced computed (CECT) scan suggesting a mass lesion in the presacral region with destruction and involvement of the underlying bone Fig. . He alsod-glucose-positron emission tomography (FDG PET) scan showed lytic lesion in the sacrum with no other areas of FDG uptake.On clinical evaluation, the patient had an antalgic gait with weight-bearing on the right lower limb. Neurological examination showed normal higher mental functions and cranial nerves. Muscle power was 3/5 at left hip, 4/5 at the left knee, and ankle joints. Power in the right lower limb joints was normal. No sensory abnormalities were identified. The straight leg-raising test on the left side was restricted to 40° and was associated with sharp pain. Superficial and deep reflexes were normal in both limbs. The patient had neither bowel and urinary complaints nor history of fever, night sweats, or weight loss. Per abdomen, per rectal, and supraclavicular fossa examination revealed no abnormality. No clinically palpable nodes on systemic examination were found. With a tentative diagnosis of bone secondaries from an unknown primary, he was planned for image-guided core needle biopsy from the sacral mass that was carried out under full aseptic precautions. The histopathology (HPE) was suggestive of malignant round cell tumors. On immunohistochemistry (IHC), cells were positive for CD20 (score 4+), CD10 (score 1+), and ki67 (score 4+). The histological diagnosis was high-grade B cell non-Hodgkin lymphoma (NHL) compatible with diffuse large B cell lymphoma . After completion of 3 weekly, 6 cycles, the patient was relieved of his pain and numbness and regained 5/5 power in the left lower limb muscles on clinical examination. Reevaluation by FDG-PET was done which showed a mild FDG avid sclerotic lesion of 1.3 × 1.2 cm in the left ala of the sacrum with standardized uptake value (SUV) of 3.22 suggesting a mild active residual disease  AND (“lymphoma”[MeSH Terms] OR “lymphoma”[All Fields] OR “lymphomas”[All Fields] OR “lymphoma s”[All Fields]). The title and abstracts of all the articles were reviewed and for articles wherein no information was provided in abstract and the article that were finally included in review full articles were extracted. Data on age, gender, histological type, treatment, follow-up, and response to treatment was extracted. Data was tabulated.A total of 139 articles were extracted using the search string. Of these, 25 articles were selected and 116 articles were excluded as they did not report on primary sacral lymphoma Fig. . After rPrimary bone lymphomas are rare constituting less than 2% of all lymphomas in the adult population. These tumors mostly involve the femur, other long bones of extremities, and pelvis , 2. PrimPrimary sacral lymphoma is very rare entity and has imaging similarities with other tumors of sacrum. Definitive diagnosis is made only by HPE, and IHC is required for differentiating it from the rest of bony lesions. Hence, this case demonstrates the importance of insisting on core biopsy and not relying only on fine needle cytology if the diagnosis does not fit the overall clinical picture; non-Hodgkin's lymphoma has a good response to the R-CHOP regimen and radiotherapy while in a metastatic disease the outcome is dismal. Although few studies show better prognosis in primary bone NHL, the incidence and prognosis of primary sacral lymphoma are not still very clear because of very few reported cases.Primary sacral lymphoma is a rare differential diagnosis for primary sacral tumor.Definitive diagnosis is by biopsy and immunohistochemistry.Treatment options are R-CHOP regimen, radiation therapy, and surgical decompression.It is important to obtain a core biopsy and do immunohistochemistry in cases where the imaging and clinical picture does not support the provisional diagnosis.Depending upon cytology alone can be disastrous at times.


Malignant SMARCB1/INI1-deficient extrarenal rhabdoid tumors are aggressive tumors that are extremely rare in adults. A 56-year-old male presented with the chief complaints of unilateral lower abdominal and pelvic pain. He underwent urgent surgical intervention and mass resection with tissue sampling. After pathology confirmed the diagnosis, systemic chemotherapy with vincristine, doxorubicin plus ifosfamide, and mesna was administered. Following treatment, he experienced a durable and long-lasting response to therapy for this aggressive and rare soft tissue sarcoma. To date, the patient remains in complete remission following the cessation of chemotherapy. Malignant SMARCB1/INI1-deficient extrarenal rhabdoid tumors are aggressive neoplasms that are extremely rare in adults. We report a rare case of such a tumor and review the literature for its molecular, clinical, and imaging features. The tumor-suppressor gene SWItch/sucrose non‐fermentable-related (SWI/SNF) matrix-associated actin-dependent regulator of chromatin subfamily B member 1 (SMARCB1)/INI1 was first described in the malignant rhabdoid tumor (MRT) of infancy . SMARCB1A 56-year-old Caucasian male presented to his primary care provider with the chief complaint of painless left lower abdominal, inguinal, and scrotal swelling. Past medical history was only notable for chronic moderate-to-heavy chewing tobacco use. Family history was notable only for smoking tobacco-associated lung and bladder cancer in his father and mother, respectively, and prostate cancer in his maternal grandfather. This symptom started six months prior to presentation and had been gradually progressing. He denied any previous abdominopelvic trauma or hernia diagnosis. Vital signs, the remainder of the physical examination, and laboratory testing were primarily benign. A detailed physical exam was remarkable to the suprapubic area. Ultrasound of the pelvis was obtained. Sonographically, fluid collections were not sizeable enough to explain his symptoms. CT scans with contrast of the abdomen and pelvis were performed and showed a large soft tissue mass (approximately 12 cm x 12 cm x 23 cm) originating from the left testicle or scrotum that extended superiorly out of the scrotum and into the inguinal canal. From there, the soft tissue mass tracked along the left side of the abdominal and oblique muscles. There was no radiologic evidence of a fat plane between the musculature which was suspicious for direct invasion. Moreover, retroperitoneal adenopathy was not evident, and there were no obvious signs of metastases in the abdominopelvic cavity. Exploratory laparoscopy with left orchiectomy and mass debulking was performed in an effort for symptom management, tumor resection, and diagnosis. Operative notes illustrated that gross tumor invasion was evident in the skin, abdominal wall, and pubic bone.Grossly, the ill-defined left inguinal/scrotal/testicular mass was 19 cm x 16 cm x 7 cm. Tumor invasion into paratesticular adipose tissue was apparent. Microscopically, sections of recognizable spermatic cord, epididymis, testis, and rete testis had no obvious evidence of primary malignancy such as a seminoma. Immunohistochemical (IHC) stains with proper controls were performed to define the tumor type. IHC stains on the malignant-appearing tissue were negative for pan-cytokeratin AE1/AE3 (pankeratin), S100, desmin, and muscle-specific actin (MSA). Tumor specimens were partially immunoreactive to CD34 and epithelial membrane antigen (EMA) markers (Figure Three months following surgical debulking, contrasted CT scans of the chest, abdomen, and pelvis were obtained, which again showed no evidence of distant metastatic disease. However, residual disease was present and acute worsening was apparent based on radiologic evidence of new rapid growth. Progressive disease was observed in the left inguinal canal measuring 7 cm x 2.9 cm, a lower abdominal wall lesion measuring 16.6 cm x 10.4 cm, and one left inguinal lymph node measuring 2.5 cm. Subsequently the case was reviewed for a second opinion at an outside facility. There, a repeat CT of the abdomen and pelvis with contrast was obtained and confirmed progressive disease. A complex nodular mass in the subcutaneous tissues of the left inguinal region and abdominal wall was observed. However, its size had approximately doubled in all dimensions. Again, no evidence of metastatic disease was present. Additional testing was performed with revealed tumor cells, showing a loss of SMARCB1/INI1 expression and were FISH-negative for NR4A3. Ultimately, the patient’s tumor was determined to be more likely a malignant SMARCB1/INI1-deficient extrarenal rhabdoid tumor.Due to the extent of disease, neoadjuvant chemotherapy was recommended. The patient underwent a regimen with vincristine, doxorubicin plus ifosfamide, and mesna. Supplemental dexrazoxane was added at the start of each chemotherapy cycle for cardioprotection. Prophylactic pegfilgrastim was given in between cycles for treatment-associated neutropenic fever and sepsis, which the patient was at an increased risk for developing on the basis of receiving myelosuppressive chemotherapy and the advanced stage of his disease.Six months after the initiation of chemotherapy, a surveillance PET/CT showed complete resolution. This is consistent with a durable and long-lasting response to chemotherapy for this aggressive and rare soft tissue sarcoma. The patient remains alive, healthy, symptom-free, and in complete remission almost 12 months following cessation of chemotherapy.Extrarenal rhabdoid sarcomas are rare soft tissue neoplasms in adults. Since being first described in 1970, its diagnosis is often difficult . Loss ofIn this case, the tumor was determined to be a high-grade neoplasm with rhabdoid morphology, most likely a malignant extrarenal rhabdoid tumor. Most rhabdoid tumors, regardless of location, are characterized by loss of expression of the SMARCB1/INI1 tumor-suppressor gene product. Keratin expression can be very limited in tissue samples. Also specimens may show expression of unusual markers, such as synaptophysin, a finding that was present in this case . Due to Malignant SMARCB1/IN1-deficient extrarenal rhabdoid tumors are a variety of rare soft tissue sarcoma in adult patients with aggressive features. Tissue biopsy is the confirmatory test of choice. However, despite clinician due diligence and the presence of supportive histocytomorphologic evidence, the diagnosis remains challenging. IHC can help elucidate the inactivation of SMARCB1/INI1 and thereby support the diagnosis of malignant extrarenal rhabdoid tumors in adults. Early identification is imperative and helps guide therapy with chemotherapy regimens such as AIM.


Desmoid-type fibromatosis (DF), also known as desmoid tumor, is an extremely rare, benign, mesenchymal fibrous tumor with no potential for metastasis. It can arise from any part of the body, most commonly extra-abdominally. Intra-abdominal DF can present sporadically, in sites of previous trauma, surgical scars and irradiation, or in association with familial adenomatous polyposis and Gardner syndrome. Intra-abdominal DF is uncommon and especially rare after a common surgery like cholecystectomy. We report a rare case of a 67-year-old male who presented with a locally aggressive intra-abdominal DF in the gallbladder fossa, status post cholecystectomy. This progressively enlarging infiltrative enhancing solid mass in the gallbladder fossa on serial computed tomography and magnetic resonance imaging demonstrated gastric outlet obstruction, biliary obstruction, portal vein narrowing and encasement of hepatic artery. Diagnosis of DF in this postoperative setting was delayed and challenging due to uncharacteristic clinical presentation. Radiologists should be aware of this unusual diagnosis and spectrum of imaging findings to help in timely surgical management and planning. Desmoid-type fibromatosis (DF) is a rare benign fibrous tumor that either presents as an indolent or locally aggressive mass. It is mostly encountered extra-abdominally, affecting the extremities, head, neck, chest and breast. Intra-abdominal DF can occasionally be seen at sites of prior trauma, scars and radiation therapy, or in association with familial adenomatous polyposis (FAP) and Gardner syndrome . LocallyA 67-year-old male complaining of persistent right upper quadrant (RUQ) pain for over 10 months, status post cholecystectomy. The pain started one month after cholecystectomy performed at an outside hospital. This pain was radiating to epigastric region and associated with nausea, vomiting and unintentional weight loss of about 30 lbs since the onset of the pain. At the time of his prior surgery, his gallbladder was found to be severely inflamed, with dense adhesions. Cholecystectomy was performed without any complications, and the patient was discharged safely.After the RUQ pain started, he underwent extensive workup at an outside hospital, which included computed tomography (CT), magnetic resonance imaging (MRI), magnetic resonance cholangiopancreatography, esophagogastroduodenoscopy and colonoscopy. Review of initial CT Figure  and MRI Diagnostic laparoscopy and biopsy were performed at the outside hospital, which revealed predominantly fibrous soft tissue on pathology. At that time, it was concluded that this was a benign process not likely to result in postoperative scarring, and the patient was discharged with anti-inflammatory medications.The patient subsequently presented at our hospital, with acute abdominal pain and symptoms of gastric outlet obstruction, leading to surgical consult. After review of his prior history and investigations, the patient underwent an exploratory laparotomy. At the time of surgery, the mass indeed appeared to cause gastric outlet obstruction and a gastrojejunostomy was performed. Complete excision was not possible due to unexpected increased size of this gallbladder fossa mass resulting in advanced infiltration to porta hepatis structures. Multiple excisional biopsies were taken for pathological analysis.The abdominal mass biopsies were stained with hematoxylin and eosin stain for histological examination. This revealed proliferative broad sweeping fascicles of spindle-shaped fibroblasts in an abundantly collagenized background. The fibroblasts were separated by thin-walled, delicate blood vessels that were aligned along edges of fascicles Figure . The tumRepeat post-operative CT performed shortly after the gastrojejunostomy demonstrated a significant interval increase in size of the mass Figure , compareThe patient’s management and pathology was discussed at the hepatobiliary multidisciplinary conference in view of failed resection. The possibility of a complete tumor resection was excluded due to extensive local invasion and involvement of multiple structures and the porta hepatis. The decision was made in favor of nonsurgical management, imaging follow-up and trial of tamoxifen therapy as suggested by the oncologist.DF, also known as aggressive fibromatosis, deep fibromatosis or desmoid tumor, is a rare sarcoma subtype that is benign and does not metastasize. It accounts for 0.03% of all neoplasms and 3% of all soft tissue neoplasms . It is uThe clinical presentation of DF is unpredictable, as it may range from being asymptomatic and indolent mass to locally aggressive mass with complications related to local obstruction and infiltration. It can arise from any mesenchymal tissue in the body, mostly seen extra-abdominally originating from musculoaponeurotic tissues . CT and MRI is the preferred imaging modality to assess extra-abdominal and intra-abdominal DF. The MRI signal intensity pattern reflects the proportion of different tissues present microscopically. It most commonly presents heterogeneously with hypo/hyperintense signal on T2 images and iso/hypointense signal on T1 images. The band sign has been demonstrated on all MRI sequences for DF, described as hypointense nonenhancing linear bands, presumably representing dense collagen stroma. Although common, it is not a pathognomonic sign, as it can be seen in other soft-tissue tumors such as giant cell tumor of tendon sheath and myxofibrosarcoma. Enhancement after administration of gadolinium contrast media is commonly seen, particularly in hypercellular regions of the tumor. The value of diffusion-weighted MRI sequences is still unclear .Although the imaging pattern will raise the possibility of DF, histological analysis is always required to rule out differential diagnosis. Immunohistochemical studies of desmoid tumors have demonstrated a fibroblastic-myofibroblastic phenotype. Muscle-specific actin, SMA and desmin are expressed in varying proportions of cases . CoexpreSurgery is the standard treatment for DF in symptomatic patients without significant morbidity. However, for high-risk patients and locally advanced infiltrative masses, complete excision could lead to a potential loss of function, and increased morbidity and mortality. Moreover, the literature is controversial regarding the prognostic significance of either positive or negative margins for DF tumors . ProgresDF is a rare entity with peculiar imaging features, resulting in delayed diagnosis. Radiologists should be aware of this unusual diagnosis and spectrum of imaging findings including locally aggressive behavior. Early tissue sampling with laparoscopic/open biopsy is advisable for timely diagnosis, surgical management and ruling out other neoplasms.


Teratomas arise from primordial germ cells which arrest during its migration from the hindgut allantois the gonads during the first weeks of gestational life, they may occur in both gonadal and extra-gonadal locations. They occur in 1/40,000 live births. The most common anatomical locations are the sacro-coccygeal region and the ovary, neck teratomas constituted about 1.5%. Malignant transformation has been reported.A 2-year-old boy presented with a gradually enlarging mass in the left side of the neck causing stridor and difficulties in respiration especially during sleep, the parents noticed difficulties during swallowing. The mass was misdiagnosed as cystic hygroma and the patient underwent 2 sessions of sclerotherapy with no improvement. Clinical examination showed a large mass in the left side of the neck which was multilobulated causing tracheal shift to the opposite side. There were no signs of inflammations over the mass. CT-scan showed evidence of enhancing multi-cystic lesion with multiple flecks of calcification. Complete surgical resection was done successfully and the histopathological study of the mass showed mature elements of ectodermal, mesodermal, and ectodermal germinal layers which was consistent with mature cystic teratoma. The follow up showed no postoperative complications.Mature cystic teratoma of the neck is very rare and may be difficult to be differentiated both clinically and radiologically from other neck masses. The accurate diagnosis must be made before any kind of treatment is started. Complete surgical resection is the main management option and required to decrease the recurrence. Due to this migration they may occur in both gonadal and extra-gonadal locations and can be localized anywhere from the head to the coccyx .The incidence of teratomas is approximately 1/40,000 live births with slight female predominance. The most common anatomical locations of teratomas are the sacro-coccygeal region and the ovary, rarely they may occur in other sites like the mediastinum and any other anatomical site, neck teratomas constituted about 1.5% of all teratomas .Histological examination show that teratomas are composed of heterogeneous with mixed cystic, solid, mature, and immature components. Histologically they are classified into mature teratomas which are benign and constituted about 95% of cases and immature teratomas which involve malignant transformation .Teratomas are usually asymptomatic and clinically vary greatly in size, they may be small in size but sometimes they may reach an enormous size causing pressure effects resulting in obstruction of the respiratory or digestive passages ,3.Imaging help to detect the size and the extension of the mass, ultrasound show the size and the composition and detect any cystic components inside the mass, CT scan and MRI are more useful to show the anatomical details and the origin of the mass .Surgery is the main management option and complete surgical resection required to decrease the rate of recurrence, malignant transformation has been reported in some cases and such cases require more extensive surgical resection. Radiotherapy and chemotherapy are usually not very effective in such cases ,3.The aim of presenting this case is to highlight the differential diagnosis of neck masses in children and the role of surgery and histopathology in the confirmation of teratomas that may be misdiagnosed and managed as cystic hygroma.The work of this report case has been reported in line with the SCARE 2020 criteria .2A 2-year-old boy presented with a gradually enlarging mass in the left side of the neck causing stridor and difficulties in respiration especially during sleep, the parents noticed difficulties during swallowing. There were negative personal or family histories for medical or other genetic disorders. The drug history was also negative.The mass was misdiagnosed as cystic hygroma of the neck and the patient underwent 2 sessions of sclerotherapy with no any clinical improvement.2.1During clinical examination the patient had a large mass in the left side of the neck, its size was 10 × 8 × 8 cm in maximum dimensions. The mass was extending to the left check superiorly, the posterior neck, the supraclavicular region inferiorly, and crossing the midline of the anterior neck. The mass was multilobulated with areas mixed soft and solid consistencies during palpation. The trans-illumination test was negative. The trachea was shifted to the opposite side. There were no signs of inflammations over the mass. The vital signs were normal and other parts of the examination were unremarkable .Fig. 1A 2.2CT-scan showed evidence of enhancing multi-cystic lesion with multiple flecks of calcification and causing displacement of the trachea, esophagus, and carotid sheath to the opposite side. There were no enlarged cervical lymph nodes .Fig. 2CT2.3The patient was admitted for surgical intervention. An oblique elliptical was done over the mass, dissection done and the mass was completely separated from the neck structures and complete surgical excision was performed. There were no signs of local invasion , Fig. 4.The operation was done by 2 general surgeons who are specialized in the field of general surgery and neck surgery.The histopathological study of the mass showed mature elements of ectodermal, mesodermal, and ectodermal germinal layers. No immature elements were seen. The appearance was consistent with mature cystic teratoma .Fig. 5Mi2.4The patient was admitted for 3 days after surgery, the drain was removed at the 2nd postoperative day. The follow up showed no postoperative complications. No specific postoperative considerations were present.3Teratomas are composed of all the three germ cell layers: ectoderm, mesoderm and endoderm that exhibit various degrees of maturity, typically they consist of mature elements of various types of tissues. Teratomas can occur in gonadal or extra-gonadal locations. Sacrococcygeal teratomas are most common in children and teratomas of the gonads are most common in adults, neck teratomas are relatively rare and usually are found as an extension from mediastinal origin ,2,5,6.They present as slowly enlarging masses unless if they developed malignant transformation, classically they exhibit solid or mixed cystic and solid components. The majority of mature teratomas contain epithelial and sebaceous components .Cystic teratomas may be difficult to be differentiated both clinically and radiologically from lymphatic vascular malformations and such patients may be treated with sclerotherapy which usually shows no response .Most teratomas contain fat which is of mesodermal origin and it is an imaging hallmark and has a very important diagnostic value during imaging. The contents of the teratomas are best assessed by CT scan. CT scan frequently assess the presence of coarse or fine linear patterns of calcium deposition. The detection of fat inside the tumor whether as sebum or macroscopic fat is the most characteristic finding of mature teratomas. Fat is demonstrated in standard imaging and typically has less than −20 HU attenuation on CT scan images and a decreased signal intensity on fat saturated MRI images .Neck teratomas must be differentiated from cystic hygroma, branchial cyst, hydatid cyst, enlarged cervical lymph node, and thyroid related pathologies. When a child present with one congenital malformation, complete clinical examination should be done to role out other possible associated malformations .Some complications have been reported in patents with mature teratomas such as infection, rupture, malignant transformation, hormone secretion which occur particularly in cases of struma ovarii, torsion in cases of ovarian teratomas. Malignant transformation is estimated to occur in 2%–3% and is more frequent in children and can originate from any of the three germinal cell layer, the majority of malignant transformations give rise to squamous cell carcinoma, and other types of cancers may include adenocarcinoma and sarcoma. Malignant transformation is suspected when there is rapid change in size, abnormal wall thickening and irregular walls, and evidence of local invasion [Most head and neck teratomas are small and amenable to curative surgical resection, sometimes patients may have delay presentation and they may reach a very large size making surgical intervention challenging. In adult patients, immature tumors are more likely to exhibit malignant behavior ,13,14.The prognosis is usually favorable providing that the correct diagnosis is made. In a case series presentation involving 23 patients with head and neck teratomas, the authors reported an excellent prognosis after surgical intervention. The presented case had been treated successfully with surgery and the follow up showed no postoperative complications and recurrence ,16.Neck masses may be misdiagnosed due to the variety of differential diagnoses, our particular case was misdiagnosed as cystic hygroma and because of no response to sclerotherapy we thought of an alternative diagnosis.The author has no conflicts of interest to declare.None.Ethical approval has been exempted by my institution for reporting this case.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.Dr Hassan Muhsen Hassan and Dr Asaad Shareef Omar contributed to the concept of reporting the case and the patient data recording.Drafting the work, design, and revision done by Dr Ayad Ahmad Mohammed.Final approval of the work to be published was done by Dr Ayad Ahmad Mohammed.This work is case report and there is no need of registration.Dr Ayad Ahmad Mohammed is guarantor for the work.Not commissioned, externally peer reviewed.The parents were hoping a complete cure for their child, after surgery and the result of histopathology they were very hopeful and happy about the results of both surgery and the final diagnosis.


Endometrial adenocarcinomas arising in a complete septate uterus with cervical and vaginal duplication are rare. Here, we report a case of stage III endometrioid endometrial adenocarcinoma arising in a complete septate uterus with a double cervix and vagina coupled with a left serous ovarian cystadenoma in a 35-year-old-female patient. The patient underwent a total abdominal hysterectomy with bilateral salpingo-oophorectomy and was addressed to the oncologist for adjuvant radio-chemotherapy. We highlight the symptoms, diagnosis, and therapeutic management, and compare them to the recent literature.  The uterus is the fourth most common primary site of cancer among women, and more than 80% of uterine cancers are endometrial adenocarcinomas . Congeni2 was referred to our hospital for complaint of dysfunctional uterine bleeding for two months. She had her menarche at the age of 14, with regular cycles, slight dysmenorrhoea, and dyspareunia. Her past medical history was noticeable for a miscarriage after eight weeks of gestation and a five-year history of type 2 diabetes under insulin therapy. There was no family history of any congenital anomalies, and the patient was unaware of her uterine anomaly. Gynecologic examinations revealed a phenotypic female (Tanner stage V), a soft abdomen with no tenderness on palpation of the bilateral adnexa, a normal with double cervices, a double vagina with a longitudinal vaginal septum, no contact bleeding, and the external genitalia was normal. We had no previous information concerning her gynecologic examination in between her miscarriage and bleeding. A 35-year-old female patient  with a body mass index (BMI) of 29.6kg/mHysteroscopy was performed with uterine endometrial biopsy, which revealed a malignant tumor, suggestive of moderately differentiated adenocarcinoma. Genetic counseling for cancer was unavailable, and ultrasound was not obtained. Abdomino-pelvic contrasted computerized tomography (CT) scan revealed a slightly increased size of the uterus, 87x37x70mm, double uterine cavities, raising the concern for stage III endometrial tumor thickening invading more than 50% of the thickness of the uterine muscle, with the onset of bilateral invasion of the parametria, and small lymph nodes without infiltrating the cervix Figure .After a complete anesthesia fitness, glycemic control with insulin, and informed consent, the patient underwent an exploratory laparotomy where a total abdominal hysterectomy, bilateral salpingo-oophorectomy, and staging workup  were performed. At the time of surgery, the unusual per-operative finding was that of a septate uterus with complete duplication of the cervix and vagina Figures -4. The patient’s postoperative course was uneventful. Gross examination revealed a complete septate uterus, with a complete septum from the fundus to cervix measuring 6x6x3.5cm and a double cervix measuring 4x2.5x3cm for the largest. Microscopic examination revealed, as designated by the International Federation of Gynecology and Obstetrics (FIGO), a grade II endometrial endometrioid adenocarcinoma, infiltrating the entire uterine wall coming into contact with the serosa, the isthmus, and the lymph nodes, with a left serous ovarian cystadenoma.2 given intravenously during radiotherapy, followed by four cycles of carboplatin and paclitaxel 175mg/m2 given intravenously. A follow-up period of eight months has shown no signs of recurrence.The disease was consistent with FIGO stage IIIC (pT1bN1Mx). She recovered rapidly from surgery and was addressed for adjuvant combined radio-chemotherapy to the pelvic and para-aortic region for high-risk endometrial cancer. She received a total dose of 46Gy of external beam radiotherapy in two fractions given on five days per week, two cycles of cisplatin 50mg/mTo discuss this rare condition, we performed a literature review of cases of endometrial carcinoma (EC) associated with Müllerian duct anomalies (MDA). Müllerian anomalies have not been implicated as a significant risk factor for the development of cervical, uterine, and ovarian cancers; however, several reports describe the existence of various tumors in anomalous uteri -7. We deA total of 27 cases of EC arising within MDA (including our case) have been reported. Of the 27 patients, the ratio of uterus didelphys was as high as 48.1% (13/27), bicornuate uterus was 33.3% 9/27), and septate uterus was 14.8% (4/27). EC arising in the septate uterus is rare, and the present report represents the fourth of such cases being reported in the literature. Itoh et al. described the first case in 2014 in a 39-year-old patient with a septate uterus who presented with postpartum bleeding. The histological diagnosis was consistent with well to moderately differentiated, endometrioid adenocarcinoma arising in both cavities of a septate uterus, which was detected six months after full-term delivery /27, and . The thiThe behavior and incidence of EC arising in a septate uterus have not been well documented and less in its association with ovarian cystadenoma. Women with MDA appear to have a higher rate of unexplained infertility, endometriosis, spontaneous abortion, breech presentation, and premature delivery , 13. ThoPelvic ultrasonography, CT scan or magnetic resonance imagery (MRI), hysteroscopy, and dilatation and curettage (D&C) or endometrial biopsy are all among the diagnostic evaluation of EC. Pelvic MRI is the investigation of choice for detection of MDA with an accuracy of up to 100% being reported . It is uAccording to the sparse literature, the simultaneous presence of primary cancers in the endometrium and the ovary is not well documented. Approximately 5% of all patients with EC appear to have ovarian cancer synchronously, and 10% the other way around . To the All of these four cases of EC arising on septate uterus underwent complete staging surgery, and this present report had advanced Tanced stage III disease, and two cases had stage I disease according to the revised FIGO staging system . They alUterine anomalies are rare and their diagnosis may go undetected. The coexistence of uterine malformation and EC poses the problem of confirmation in some situations and sometimes the diagnosis is made intraoperatively. Surgeons should not be surprised when they come across this unusual condition.


The lacrimal ductal cyst (dacryops) is an uncommon clinical entity. It occurs anywhere that lacrimal gland tissue is present but most often appears as an expanding mass in the region of the lacrimal gland. The presence involving the medial part of the orbit is rare, ectopic location can be misleading in the differential diagnosis of orbital masses. The authors report a 53-year-old man who presented with dacryops occurred in an unusual location with significant clinical presentations.A 53-year-old man had a painless mass located in the right superomedial orbit accompanied with foreign body sensation and lachrymation for two months, which had rapidly grown within 10 days. Decrease of visual acuity, high intraocular pressure (IOP) and limitation of extraocular movements in the right eye were present. The result of visual evoked potential (VEP) test suggested the impaired function of the optic nerve. Magnetic resonance imaging (MRI) studies revealed the presence of an isolated cystic lesion. The mass was completely removed via a transcutaneous approach, histopathologic findings were consistent with the lacrimal ductal cyst. The ocular motility and high IOP returned to normal. There had been no post-operative complications or signs of recurrence over five months follow-up.Lacrimal ductal cysts can present in the medial orbit, clinicians should include this entity in the differential diagnosis of orbital masses and be aware of its variable presentations such as high IOP in this case. We comment on the fact that many reported cases of ectopic dacryops may be an extension of normal lacrimal gland tissue. The lacrimal ductal cyst (dacryops) is a benign epithelial tumor of the lacrimal tissue, which usually locates in the outer part of the upper eyelid presenting to be an asymptomatic, smooth, mobile and fluctuant swelling . EctopicA 53-year-old man complained of a painless swelling of the right upper lid for 2 months accompanied with foreign body sensation and lachrymation. The mass had rapidly developed within 10 days, which could be enlarged and purplish blue after rubbing the eye. The patient denied the history of trauma, infection or surgery. A cystic mass could be felt at the superior-medial of the orbit, which was smooth, mobile, fluctuant and without tenderness. There was some underactive motility when the patient looked upward (Fig. Lacrimal ductal cyst is not common, from studies in Wills Eye Hospital by Shields et al.,it accounts for up to 2% of all orbital mass and 6% of lacrimal gland lesions , 8. The Among all forms of dacryops from the literature, simple dacryops is the most common which often protrude into the lateral upper fornix , 9, 10. The pathogenesis for the development of dacryops is still unclear. It has been reported associated with trachoma, conjunctival inflammation, ocular cicatricial pemphigoid, high levels of IgA, prolapse of the lacrimal gland, ocular trauma and chemical injury , 15, 18.Dacryops is usually asymptomatic and insidious, which can grow rapidly in the context of infection .. The chIn conclusion, we describe an uncommon lacrimal ductal cyst in an atypical location, which should be considered in the differential diagnosis of medial orbital masses. Although the dacryops presents as benign tumor, the mass effects deserve attention for treatment.


Intestinal endometriosis is rare and most frequently involves the rectum and sigmoid colon.Here, we report a case series of 5 patients who underwent laparoscopic resection for intestinal endometriosis. None of the patients developed postoperative complications, and all were discharged at 5–8 days after surgery. The diagnosis of intestinal endometriosis is difficult to obtain before surgery. Only 2 of 5 patients were diagnosed preoperatively. Among 1 of the 2 patients, the symptoms at the time of menstruation were obvious. In patients with submucosal tumors, the preoperative diagnosis can be difficult. Additional imaging examinations at the time of menstruation might be useful for obtaining a diagnosis. D2 dissections were performed for 3 patients, because malignancy could not be ruled out as a preoperative diagnosis. The surgical findings of 1 patient did not appear to be endometriosis. Surgery for intestinal endometriosis usually encounters advanced pelvic adhesions and fibrosis. For patients undergoing sigmoidectomy, the mean operative time was 152 min and mean blood loss was 10 mL. For patients undergoing rectal resection, the mean operative time was 282 min and mean blood loss was 17 mL. Two cases had severe pelvic adhesions, and the residual rectum could not be straightened. Therefore, side-to-side anastomosis was performed. For intestinal endometriosis surgery, flexible planning for the anastomosis method used for residual intestine should be undertaken.Laparoscopic surgery for intestinal endometriosis was safe but technically difficult, because of fibrosis and adhesions. An accurate diagnosis should be attempted based on the clinical symptoms, imaging findings, and intraoperative findings. The method used for anastomosis should be decided on a case-by-case basis. Intestinal endometriosis is rare. It is seen in about 10 to 30% of patients with endometriosis . Here, wAfter a positive fecal occult blood test (FOBT), colonoscopy revealed an extramural mass in the sigmoid colon Fig. , and theAfter a positive FOBT, and with a history of constipation, the patient underwent rectal examination, which revealed a palpable mass. A barium enema examination revealed a 40-mm extramural mass in the rectum Fig. , and theThe patient presented at another hospital with a history of 2 to 3 months of abdominal pain. Abdominal ultrasonography revealed a mass at the end of the ileum, and she was referred to our hospital. A colonoscopy revealed a submucosal tumor at the end of the ileum, and we marked the location by a tattoo. CT was unremarkable, but MRI revealed a mass at the end of the ileum Fig. . Our preAfter a positive FOBT, colonoscopy revealed a 25-mm submucosal tumor in the sigmoid colon Fig. , and theThe patient presented at another hospital with a 5-year history of constipation. A barium enema examination revealed an extramural mass in the sigmoid colon Fig. , and shePreoperative biopsies were performed in two of the five patients, but none were diagnosed because they were biopsies from the mucosal surface. Although endoscopic ultrasound-guided fine needle aspiration (EUS-FNA) was considered in all cases, all cases were symptomatic and early surgery was requested, so EUS-FNA was not performed.None of the patients developed postoperative complications, and all patients were discharged at 5–8 days after surgery.Endometriosis is a general term for endometrial tissue found outside the uterus and is present in 5 to 15% of women of reproductive age . It has Before surgery, intestinal endometriosis was diagnosed in 2 of the 5 patients. Otherwise, it was difficult to diagnose preoperatively. Among the 2 patients, the clarifying symptom of pain was present at the time of menstruation in only 1 patient.In case 3, non-menstrual CT showed no findings at the terminal ileum, but MRI performed during menstruation showed a mass. Because of the different modalities, it is not possible to conclude based on this report alone, but it was also important to examine the difference between menstrual and non-menstrual images in diagnostic imaging of intestinal endometriosis. Although no reports on menstrual and non-menstrual imaging have been found in previous reports, the accumulation of future cases is considered important. Obtaining a preoperative diagnosis appears to be difficult for patients with a submucosal lesion; however, Oliveira et al. reported that intestinal endometriosis should be considered in the differential diagnosis of patients with submucosal tumors of the colon . If inteD2 dissection was performed for case nos. 1, 2, and 4; because malignancy could not be ruled out preoperatively. Tumors arising from intestinal endometriosis are rare and are collectively referred to as endometriosis-associated intestinal tumor (EAIT) . MalignaThere have been reports that endometrial tissue has also been found in regional lymph nodes at the time of the resection of the intestine involved with endometriosis; however, the relationship between lymph nodes containing endometrial lesions and the malignant transformation of endometrial lesions in the intestine has not been clarified, and carcinomas associated with intestinal endometriosis are rare , 10. SomThe surgical findings of 1 of our 5 cases did not suggest endometriosis. The surgical findings of surgery for intestinal endometriosis often include advanced pelvic adhesions and fibrosis. These findings are thought to be caused by endometrial tissue, which undergoes fibrosis, and metaplasia of smooth muscle extending throughout the muscular layer to the subserosal layer of the intestinal tract and repeatedly causing bleeding and inflammation along with the menstrual cycle .Rocha et al. have evaluated laparoscopic surgery in endometriosis surgery with colectomy and report that it is also effective in alleviating abdominal symptoms and preserving fertility . For patLaparoscopic surgery for intestinal endometriosis was safe but technically difficult, because of fibrosis and adhesions. An accurate diagnosis should be attempted based on the clinical symptoms, imaging findings, and intraoperative findings. The method used for anastomosis should be decided on a case-by-case basis.


Congenital bands are rare causes of intestinal obstruction and often leads to diagnostic challenges. Diagnostic delays in cases of mechanical obstruction might lead to irreversible bowel ischemia and perforation. Presently described is a case of an 18 month young child with severe vomiting developed for one day. The child was initially thought to have acute viral enteritis and treated accordingly. Due to the severity, an X-Ray and computed tomography scan were sent which pointed towards the possibility of having congenital bands. He was treated operatively. The child was kept under observation for eleven days and was discharged. Although rare, intestinal obstruction due to congenital bands must be considered when treating a child with severe vomiting. The vomitus was bilious, projectile in nature and occurred after every meal. According to his father, the child had had his dinner the preceding night. After a few hours of sleep, the child started vomiting and was then brought to the hospital the following morning when it didn't stop throughout the night. Ondansetron 1ml was immediately given to the child orally. An intravenous line was opened and 200ml of normal saline was infused. Paediatric consultation was done and the child was admitted and shifted to the paediatric ward.The child was managed with Ondansetron and fluid replacement which decreased the frequency of vomiting. He was treated as a case of acute viral enteritis. But, considering the severity of the vomiting and the high number of episodes with which he presented, and because of the concern of the parents, a plain abdominal radiograph was sent. The X-Ray findings changed the diagnosis as well as the course of treatment entirely because it showed dilated loops of the small bowel with multiple air-fluid levels . A nasogA surgery referral was made and he underwent surgery. During which, malrotation of the gut was noted. The ileal segment was narrowed. Congenital bands were seen 35cm proximal to the ileocecal junction and were transected. There was no ischemia or peritonitis. The child was put under postoperative observation in the high care unit for 11 days.During his postoperative period, he gradually recovered and tolerated oral feed after 9 days of stay at the high care unit. He was shifted to the ward for 2 days and discharged with prescription of syrup Flexon 5ml per oral three times a day for three days.We reported a rare case of intestinal obstruction in a child, which was strongly evoked by history, and radiological findings. Although initially thought to be a viral enteritis, radiologic investigations changed the course of treatment and outcome for the patient. Had a diagnosis not been made on time, or had the patient been discharged after symptomatic relief by antiemetics, possible severe complications of intestinal obstruction could have caused the patient to land up in a worse condition.5 Although we didn't make a diagnosis of congenital bands via CECT, we did suspect it to be the cause. However, a definitive diagnosis was made operatively. Laparoscopy was also rarely used in the reported cases.5 But, laparoscopic surgery would have allowed a relatively safer and effective treatment.4In the reported cases, the diagnosis of congenital bands was made intraoperatively.6We have concluded that mechanical obstruction due to congenital bands must also be considered as a differential when children present with acute onset of excessive vomiting. A plain radiograph of the abdomen must be sent whenever in doubt and CECT must be performed to establish a diagnosis. Laparoscopy would have been a feasible, safe and effective treatment for obstructions caused by congenital bands.JNMA Case Report Consent Form was signed by the patient and the original article is attached with the patient's chart.None.


Gastric cancer that mimics a submucosal tumor (SMT) is infrequently encountered in routine clinical settings, and histopathological analysis is often negative for malignant cells. In such cases, excisional biopsy of the entire tumor may be necessary to make a definitive pathological diagnosis, and laparoscopic and endoscopic cooperative surgery (LECS) is a viable method of excisional biopsy.An 80-year-old male patient diagnosed with stomach wall irregularities at routine medical check-up was referred to our facility, and consequent endoscopic examination detected a 20-mm protruded lesion in the greater curvature at the middle third of the stomach. Endoscopic ultrasound (EUS) showed a thick, low echoic lesion with an irregular margin in the second layer of the gastric wall. Further, a nodular part of the lesion had infiltrated into the submucosa, with an appearance similar to that of linitis plastica of the stomach. The lesion was highly suspected to be a gastric carcinoma with submucosal invasion. However, mucosal-incision-assisted biopsy revealed no malignant cells. Computed tomography (CT) identified no metastatic lymph nodes. Therefore, an excisional biopsy using LECS was performed, and to avoid peritoneal dissemination, we used a modified version, namely, combination of laparoscopic and endoscopic approaches to neoplasia with non-exposure technique (CLEAN-NET). The procedure ended without any complications, and post-operative course was uneventful. As histopathology returned a diagnosis of adenocarcinoma pT4a, we performed radical gastrectomy and D2 lymphadenectomy. Post-operative course was unremarkable and the patient underwent follow-up examinations without adjuvant chemotherapy because of old age.Local resection using LECS for gastric tumors with a high suspicion of malignancy is useful and feasible. LECS could be used in similar cases. Laparoscopic and endoscopic cooperative surgery (LECS) is a procedure that combines laparoscopic gastric resection and endoscopic submucosal dissection (ESD) with appropriate but minimal surgical resection. LECS is routinely performed for local resection of gastric submucosal tumors (SMTs), including gastrointestinal stromal tumors (GIST) . In intrAn 80-year-old male patient who was found to have stomach wall irregularities during routine medical check-up was referred to Keio University Hospital. Endoscopic examination detected a 20-mm protruding lesion in the greater curvature at the middle third of the stomach Fig. . The tumTo avoid peritoneal dissemination, we chose the modified CLEAN-NET procedure . Since tThe technical details of this surgery involve (1) detecting the lesion and performing an endoscopic submucosal injection, (2) performing seromuscular dissection around the tumor under laparoscopic view, 3) cutting the mucosal layer using a mechanical stapler , Ly1a, V0, negative lateral margins , aortic stenosis, and coronary stenosis .The third scenario entails combining LECS with sentinel node navigation surgery (SNNS). Takeuchi et al. demonstrated the feasibility and accuracy of sentinel node biopsy in early gastric cancer . The senThe fourth indication is a tumor requiring diagnostic resection because of high suspicion of malignancy, e.g., gastric cancer mimicking SMT, similar to the case described here. Gastric cancers resembling SMT are very rare because they are epithelial neoplasms that originate in the lamina propria and are usually identified based on mucosal changes. Nonetheless, these tumors sometimes macroscopically mimic SMTs  and repoTotal excisional biopsy using a modified LECS procedure for a gastric tumor with a high suspicion of malignancy appears to be useful and may represent a possible treatment option for similar tumors.