Primary small bowel tumors are uncommon and usually carry a poor prognosis. Adenocarcinoma is the predominant histological type while lymphoma is far less common. Small bowel diffuse large B-cell lymphoma (DLBCL) can mimic adenocarcinoma; thus, the distinction can be challenging before tissue examination is performed. Bowel obstruction, as well as peritoneal involvement, are often seen in gastrointestinal (GI) adenocarcinoma cases; however, these features are extremely uncommon with GI lymphomas. Herein, the authors report an unusual case of an obstructing duodenal mass with peritoneal involvement, which was highly suspicious for an advanced duodenal adenocarcinoma. Surprisingly, sampling of the tumor revealed a diffuse large B-cell lymphoma. Primary small bowel neoplasms are rare tumors with an overall poor prognosis. The predominant histological type is adenocarcinoma. Other less common histologies include carcinoid, lymphoma, and sarcoma -2. TheseDespite accounting for up to 40% of cases of extranodal non-Hodgkin's lymphoma (NHL), gastrointestinal (GI) lymphoma is considered an uncommon disease . DiffuseClinically, a wide range of nonspecific symptoms can be associated with small bowel lymphoma, with abdominal pain being the most common . While bIn this report, the authors present an unusual case of small bowel obstruction secondary to a duodenal mass with accompanying peritoneal involvement. Surprisingly, the diagnosis was not adenocarcinoma, but DLBCL.A 40-year-old Hispanic woman was admitted to University Medical Center New Orleans, Louisiana in August 2017 for evaluation of progressively worsening abdominal pain of eight months duration with associated intermittent nausea and vomiting. The patient also reported losing approximately 30 kilograms in weight. She denied any associated fever, night sweats, change in bowel habits, rash, enlarged lumps or palpable masses. She was previously healthy and denied any personal or family history of malignancy. She never smoked and used alcohol sparingly. She had no known allergies.9/L. Platelet count was normal at 299 × 109/L. A peripheral blood smear was within normal limits. A chest radiograph was unremarkable. A non-contrast computed tomography (CT) scan of the abdomen revealed fluid-filled distention of the stomach and proximal duodenum with concern for proximal small bowel obstruction.On physical exam, the patient’s body temperature was 98.6°F, blood pressure was 100/60 mmHg, and heart rate was 120/min. Cardiovascular and pulmonary exams were unremarkable. The abdomen was soft and mildly tender. A palpable mass was appreciated in the right periumbilical region. No cervical, axillary or inguinal lymphadenopathy was appreciated. Laboratory analysis showed severe metabolic derangements as follows: sodium of 121 mEq/L , potassium of 2.2 mEq/L , blood urea nitrogen of 61 mg/dL , and serum creatinine of 3.03 mg/dL . Liver and pancreatic enzymes were in the normal limits. The hematologic panel was consistent with mild normocytic anemia with a hemoglobin of 11.2 g/dL along with leukocytosis with a white blood cell count of 16.6 × 10The patient was admitted to the intensive care unit given her various metabolic disturbances and was treated conservatively for partial small bowel obstruction. After optimization of her kidney function, a repeat scan of the abdomen with contrast was performed. An irregular mass-like thickening at the level of the second/third portion of the duodenum was identified resulting in severe luminal narrowing. Also, peritoneal "carcinomatosis" with multiple target lesions was noted Figure . The oveAn upper endoscopy was performed the following day revealing a frond-like friable obstructing mass in the second portion of the duodenum of which biopsies were taken Figure . Large-sImmunohistochemical stains demonstrated that these cells were strongly and diffusely positive for cluster of differentiation (CD)20 and CD79a, and negative for CD3, CD5, CD10, CD30, B-cell lymphoma 2 (Bcl-2) and pan-cytokeratin. Additionally, nuclear staining in about 40% of the neoplastic cells was present for c-Myc and in more than 30% of cells for Bcl-6. A diagnosis of DLBCL with germinal center immunophenotype was made . The Ki-67 index was elevated within the B cell population at 60%-70%. No assay specific abnormalities were detected by Bcl-2, Bcl-6, and c-Myc fluorescence in situ hybridization (FISH) probes. Completion staging with neck, chest, and pelvic CT scans failed to demonstrate any additional evidence of disease. A bone marrow examination showed no involvement by lymphoma. Human immunodeficiency virus (HIV) and hepatitis serologies were negative. Serum lactate dehydrogenase was mildly elevated at 318 U/L. Given the patient’s peritoneal lymphomatosis and weight loss, she was diagnosed with stage IV B lymphoma by Ann Arbor staging.Given the aggressive presentation with evidence of bowel obstruction and peritoneal involvement , the decision was made to start treatment as an inpatient. Therefore, cycle 1 of rituximab, cyclophosphamide, doxorubicin, vincristine, and prednisone (R-CHOP) was administered, and the patient tolerated the treatment well. Central nervous system (CNS) analysis and chemoprophylaxis were not performed as she was considered intermediate-risk for CNS involvement. A few days after the completion of cycle 1, she was able to tolerate oral feeding and she was discharged home. She received the remainder of her treatment as an outpatient and completed a total of six cycles of systemic chemotherapy. She exhibited significant clinical and radiographic improvement throughout her treatment. In fact, a repeat scan after six cycles showed improved luminal duodenal narrowing with resolution of the peritoneal deposits Figure . A push Primary small bowel neoplasms are rare tumors with an overall poor prognosis. Lymphomas are found in only 20% of small bowel neoplasms and tend to carry a better prognosis than small bowel adenocarcinomas . HoweverThe GI tract is the predominant site of extranodal NHL accounting for up to 40% of cases . ApproxiA wide range of nonspecific clinical symptoms has been reported with intestinal lymphomas. Abdominal pain is the main presenting symptom, and B symptoms are not commonly seen . Bowel oPeritoneal involvement can be seen with cases of GI adenocarcinoma, known as peritoneal carcinomatosis. Peritoneal involvement by lymphoma, known as lymphomatosis, is extremely uncommon -9. TheseDue to the low incidence of intestinal DLBCL, the optimal treatment remains unknown. Options in the form of surgery alone, surgery followed by systemic chemotherapy, chemotherapy alone, or radiation alone have been reported in the literature ,13. SurgDLBCL presenting as a duodenal mass and resulting in small bowel obstruction is a rare occurrence. Moreover, peritoneal involvement by lymphoma is extremely uncommon and remains a major diagnostic challenge to physicians. Therefore, despite being less common than adenocarcinoma, lymphoma should always remain in the differential diagnosis of GI tumors with or without peritoneal spread.


Objective. To report a rare presentation of solitary retinal capillary hemangioma manifesting with combined retinal detachment as initial presentation and its successful management.Methods. A 35-year-old healthy Indian male presented with combined retinal detachment associated with solitary retinal capillary hemangioma as initial presentation; a clinical entity still not reported in literature. Patient was managed with pars plana vitrectomy combined with retinectomy, endolaser, & silicon oil tamponade with good visual & anatomical recovery.Results. Patient had good clinical outcome with final best-corrected visual acuity (BCVA) of 6/ 24 and well attached retina at last follow-up.Conclusion. Solitary retinal capillary hemangiomas can rarely present with advanced vitreo-retinal complications like combined retinal detachment as initial manifestation that can be effectively managed with skilled & appropriate surgical intervention.  Retinal hemangiomas are mainly asymptomatic but can present with vision loss that occurs due to exudation at the macular region or secondary tractional retinal detachment, which occurs due to development of gliotic tissue involving the macula [4]. Different treatment modalities undertaken for treatment of retinal hemangiomas include laser photocoagulation, cryotherapy, photodynamic therapy (PDT), anti-VEGF, and vitreo-retinal surgery depending on clinical presentation with variable outcome in these patients [5-9]. Retinal capillary hemangiomas are benign vascular tumors of the retina either occurring as isolated tumors or in association with tumors in other systems, especially central nervous system hemangioblastomas and renal cell carcinomas as part of von Hippel-Lindau (VHL) disease [We report a case of solitary retinal hemangioma that manifested with combined retinal detachment, a rare presenting feature of retinal hemangioma, and its successful management. To the best of our knowledge, this rare presentation has not been reported in published literature until now.Fig. 1) and examination of the left eye fundus was normal. We initially considered the differential diagnosis of retinal hemangioma and vasoproliferative tumors of the retina (VPRT). Fundus fluorescein angiography confirmed the diagnosis of retinal capillary hemangioma, showing typical feeder vessel with early hyperfluorescence of vascular mass and draining vein  in the right eye, with no evidence of hemangioma in the left eye.A 35-year-old, serving military soldier presented with diminution of vision in his right eye for the past 3 months, which has rapidly worsened over the last month. His systemic parameters were within normal limits. Ocular examination revealed a best-corrected visual acuity (BCVA) of hand movement close to face (HMCF) with accurate projection of rays in the right eye and 6/ 6 in the left eye. Examination of the anterior segment was normal in both eyes. Fundus examination in the right eye revealed total retinal detachment with about 6 Disc Diameter (DD) size reddish orange mass with two large stretch breaks stretching in the inferotemporal quadrant with surrounding fibrous tractions , thus showing a successful outcome in unusual presentation. Since the patient had already reached a stage in which the treatment modalities like laser photocoagulation, cryotherapy, photodynamic therapy (PDT), anti-VEGF, did not have any role due to total retinal detachment, he underwent combined surgery with sclera band buckle (240 size) with pars plana vitrectomy and endoresection of tumor with high viscosity silicon oil tamponade. There were no intra-operative complications with stable course post surgery and attached retina. Silicon oil was removed 5 months after the initial surgery, as soon as the emulsification was noted. At 9 months post surgery, BCVA in right eye had improved to 6/ 24 with attached retina, no evidence of remnant of tumor/ reproliferation and healthy well laser retinectomy site in temporal periphery  with pars plana vitrectomy, endoresection of tumor with high viscosity silicon oil tamponade. Patient had stable post-operative period with good visual recovery. He underwent silicon oil removal 5 months post initial surgery with good anatomical and visual outcome and no evidence of tumor proliferation/ redetachment at last follow-up, which was 9 months post vitrectomy.Thus, combined retinal detachment can be an unusual or atypical presentation of rare etiology like retinal capillary hemangiomas but appropriate and skilled vitreo-retinal surgical intervention can lead to successful visual and anatomical outcomes in these patients. Availability of Data & materialFreely available on request.Competing InterestNil.Conflict of InterestNo conflict of interest was declared by the authors.Financial DisclosureThe authors declared that this study received no financial support.AcknowledgementNil.


Purpose: to highlight the diagnostic and therapeutic challenges in a case with central and peripheral corneal lesions.Methods: We present the preoperative investigations: anterior segment optical coherence tomography and ultrasound biomicroscopy;The excised cornea has been examined histopathologically.Results: Preoperative assessment showed that peripheral lesion was not ectatic;Histopathological examination points to a possible corneal degeneration, with posttraumatic or infectious etiology for the peripheral lesion and gives some reasons to sustain the diagnosis of Schnyder’s dystrophy for the central lesion.Conclusions: The association of different corneal lesions may need many investigations to establish the diagnosis and choose the most appropriate therapeutic solution. We present the case of a 48-year-old male patient, referred to our clinic for bilateral decreased visual acuity, more pronounced on right eye.History of the eye disease started at the age of 15; then the visual acuity decreased; at the age of 20 the patient got his driving license; the visual acuity was the same in both eyes, but he was not able to distinguish the very small rows on the eye chart. Eight years before he described an incident when the right eye was very red and painful, however, the visual acuity did not modify significantly at that time. The treatment consisted in different eye drops, but the patient had no medical records and could not specify the treatment. He noticed a decrease in visual acuity of both eyes in the last year, more pronounced on the right eye.Ophthalmological examination:Visual acuity:O.D. – counts fingers at 1 feetO.S. – 0,1Refraction – cannot be performedIntraocular pressure:O.D. – 14 mm Hg.O.S. – 13 mm Hg.Biomicroscopy:O.D.The corneal epithelium was intact, fluorescein staining showed no lesion. A discoid corneal opacity was located centrally, predominantly in the anterior half of the stroma, with polychromatic crystals, particularly in the centre of the lesion. The diameter of the lesion was around 5 mm. The cornea surrounding the lesion was clearly 2 millimeters from the lesion, except for the upper side, where the transparency was only for 1 millimeter. A corneal arcus was noticed nasally, temporally and inferiorly. 0,5 millimeter of clear cornea remained near the limbus.Fig. 1).Another lesion was noticed superiorly, between 10 and 14 o’clock. It started from the limbus to the centre of the cornea. The overlying epithelium was intact. Cornea seemed thinner than normal and it was opaque in all layers. Corneal neovascularization started from the limbus to the edge of the lesion, that was located approximately 3 millimeters from the limbus .A discoid opacity was located in the centre of the cornea. The dimension was the same as in the right eye, but the polychromatic crystals were less obvious. The surrounding cornea was clear. There was a corneal arcus in the periphery, on the entire corneal circumference .Ultrasound biomicroscopy was not very helpful. There was a central corneal hyperreflectivity, with an almost equal intensity in all layers and the peripheral stroma was less reflective. This might have been related to the central stromal opacity, but in ultrasound biomicroscopy images, cornea was more reflective in its central part, so that aspect could be normal .What was more important, the peripheral cornea did not show a significant thinning in the area of degeneration; on the contrary, the thickness was higher in that specific zone than in the centre of the cornea – as in a normal cornea .Optical coherence tomography of the anterior segment was more useful. A section at the level of central lesion showed a normal epithelium and a central hyperreflectivity, more pronounced in the anterior half of the stroma .Another section, realized at the level of the clear cornea between the central and peripheral lesion showed a normal cornea in the centre of the image and some changes in the periphery of the image, where the section crossed through the peripheral lesion. Fortunately, the thickness of peripheral cornea did not seem diminished .The last section, at the level of peripheral lesion, showed a normal epithelium above the lesion. The structure of the cornea alternated reflective areas with less reflective areas in a random distribution. The thickness of the peripheral cornea was normal  Peripheral corneal degenerationO.S. Stromal corneal dystrophy (possible Schnyder’s type)Differential diagnosisFor the central lesion:1.Bietti crystalline corneal dystrophy has lesions predominantly in the perilimbal peripheral stroma. Moreover, there are peripheral retinal changes with impaired night vision (absent in our patient).2.Corneal deposits from severe dysproteinemia, multiple myeloma, Waldenstrom’s macroglobulinemia, benign monoclonal gammopathy – there are no laboratory findings related to these diseases.3.Other stromal dystrophies: granular corneal dystrophy type I Groenouw, macular corneal dystrophy type II Groenouw and lattice dystrophy show different biomicroscopic changes in cornea.For the peripheral lesion1.autoimmune diseases ulcers: rheumatoid arthritis, polyarteritis nodosa – there are no clinical and laboratory findings for these diseases.2.marginal keratitis – a clear area remains between the ulcer and the limbus.3.phlyctenulosis – less extensive in surface.4.Mooren’s ulcer – is characterized by important thinning of the cornea.5.pellucid marginal degeneration – is located most frequently inferiorly.6.Terrien’s marginal degeneration – is located superiorly, as in our patient, but it induces a significant astigmatism and is bilateral, although there are asymmetrical cases.TreatmentThe patient underwent a penetrating keratoplasty. The surgery had a normal evolution, with no intraoperative incidents.Postoperative assessment for days after surgery: visual acuity was 0,2, clear donor cornea, clear lens, fundus examination with normal appearance.Fig. 8).Three months after surgery, the visual acuity was 0,4, with clear donor cornea .- Lipid deposits in the stroma at different levels, predominant anterior, but also in posterior stroma - Lipid accumulation throughout the stroma Fig. 12,13).The histologic examination of the peripheral lesion showed a normal structure of the epithelium; at the level of the stroma, there were macrophages with intense colored nuclei, hyaline transformation in the anterior part of the stroma and small areas of neovascularization. In the left part of the image, there was a normal aspect of the cornea . The aspect was compatible with an inflammatory reaction that was healed or almost healed – macrophages may be a sigh of a chronic low level inflammation in that region  dystrophy: a well-defined corneal dystrophy in which the gene has been mapped and identified and the mutations are known .The pathologist needed to be informed that we wanted to identify lipids and cholesterol in the cornea specimen, because the preparation of the piece was different. Organic solvents and resins routinely used for examining the pieces can dissolve most of the fats, so that special lipid stain, as oil red O or Sudan black, are necessary to identify the lipids in the cornea [There were also some problems related to the surgery. The suture was performed in the region of the upper peripheral degeneration. We did not know the exact nature of that lesion. It seemed to be cicatricial, non-active, but it was difficult to foresee the outcome, the reaction of that area to the sutures . Another important factor difficult to assess was postoperative astigmatism, which could be induced by a suture in that area.The association of different corneal lesions may need many investigations to establish the diagnosis and choose the most appropriate therapeutic solution. In our case, we found out that the thickness of the cornea was quite normal in the area of the peripheral degeneration, so we performed a penetrating keratoplasty with a very good result.


Splenic cord capillary hemangioma is a rare benign vascular lesion classified as a splenic hamartoma. On the other hand, non-islet cell tumor hypoglycemia (NICTH) is one of the rare causes of spontaneous hypoglycemia and is considered to be one of the paraneoplastic syndromes. To the best of our knowledge, this is the first reported case of a splenic cord capillary hemangioma with NICTH.A 25-year-old male was referred to our hospital with hypoglycemia. Except for his low blood sugar, there were no abnormal findings from laboratory tests, which included an endocrinological examination. Enhanced computed tomography confirmed the presence of a solid mass measuring about 6 cm in the retroperitoneum, and a tumorectomy was performed. During this operation, it became clear that the tumor turned out to be a splenic parenchyma, and as a result, a total splenectomy was performed. Microscopically, we diagnosed this as a cord capillary hemangioma, and through immunohistochemistry, we found that some tumor cells were positive for insulin-like growth factor -II. Fortunately, the hypoglycemia-related symptoms disappeared after surgical resection was performed. The patient is still alive and well without evidence of local tumor recurrence 15 years after the operation.Splenic cord capillary hemangioma, one of the types of splenic hamartomas, is a very rare benign vascular lesion and might be associated with hypoglycemia thought to be NICTH. Splenic cord capillary hemangioma is a rare benign vascular lesion. Once classified under splenic hamartoma, cord capillary hemangioma is now differentiated from hamartoma, as its clonality has recently been proven . And it This patient was a 25-year-old male who presented with pale skin and sweating to our hospital. His blood glucose level was found to be in the range of hypoglycemia (52 mg/dL), and his symptoms resolved after intravenous dextrose administration. He had experienced this type of episode twice before he visited our hospital but did not have any problems during the previous few days. He had no family history of any endocrine metabolic diseases, and physical examinations showed no special findings. The patient was admitted on the basis of hypoglycemia for 2 weeks. Except for the blood sugar episode, laboratory results regarding complete blood count, routine blood chemistry, kidney function tests, urine analysis, and chest radiograph were all within normal limits. Fasting and oral glucose tolerance load tests were performed, and there were no evidences of the irregular secretion of insulin. Adding to the puzzle, blood glucose levels were within normal limits in the hospital.However, enhanced computed tomography (CT) confirmed the presence of a solid mass measuring about 6 cm with well-defined and regular borders in the retroperitoneum. Peak contrast enhancement in a part of the tumor was seen in the delayed phase. The mass seemed to arise from the adrenal gland or kidney, and there were no metastatic lesions Fig. . An MRI Hypoglycemia-related symptoms disappeared after surgical resection was performed. The patient is still alive and well without evidence of local tumor recurrence 15 years after the operation.Grossly, the resected spleen (130 g: 10 × 6.5 × 4 cm) was received. The solid tumor (3.5 × 2.7 × 2.5 cm) was encapsulated by spleen parenchyma, and the cut surface was a yellowish white  and the cortisol level was 16.6 μg/dL (4.0–23.3 μg/dL) . MoreoveWe reported a case of a splenic cord capillary hemangioma with NICTH. This is especially noteworthy since cases of splenic cord capillary hemangioma are extremely rare and more so in combination with NICTH. Because of the hypoglycemia-related symptoms in this case, the tumor was found in the early stage. For pathologists, careful histological examination is necessary not only to make a correct diagnosis with splenic cord capillary hemangioma but also to evaluate the presence of the high molecular weight form of IGF-II when the patients have hypoglycemia-related symptoms and consider the possibility of NITCH.


Blue rubber bleb nevus syndrome (BRBNS) is a rare disease in which venous malformations (VMs) involve any body organ, most commonly the skin and the gastrointestinal (GI) tract. Treatment of BRBNS aims at preserving the GI tract as much as possible. Although there are several dozen case reports about BRBNS that describe short-term clinical courses, a few provide an account of long-term clinical course. Here, we report a case of BRBNS in a girl that required multiple abdominal surgeries due to the GI VMs and a recurrence at an interval of 14 years. The preferred approach for gastrointestinal VMs involves the complete resection of all lesions without residual VMs. It is important to bear in mind the possibility of delayed recurrence of GI VMs after surgical or endoscopic treatment. Blue rubber bleb nevus syndrome (BRBNS) is a rare condition, characterized by multiple venous malformations (VMs) of the skin and gastrointestinal (GI) tract , 2. The We report the case of BRBNS with GI lesions involving long-term follow-up and multiple surgical treatments. We also provide a brief review of the literature regarding long-term follow-up of GI lesions of BRBNS treated endoscopically or surgically.A 19-year-old girl was admitted to our hospital for surgery with a 15-year history of GI bleeding associated with BRBNS. She had repeated episodes of resections of hemangiolymphangiomas on the right thoracic wall from 3 months to 3 years of age. At 4 years of age, she presented with a few cutaneous small VMs on the feet; hematological examination revealed persistent anemia and positive fecal occult blood. Colonoscopy revealed a VM on the right colon. She underwent an exploratory laparotomy at 5 years of age. Multiple VMs were identified : 1 on thβ-blocker since she was 18. Hematological examination revealed that Hb level was 4 mg/dL. Frequent blood transfusion was prescribed. At 19 years of age, CT revealed an increase in the size of known lesions. In order to control GI bleeding, she underwent the second surgery for GI VMs. We performed resection of all lesions on the stomach and the intestine, partial resection of the small intestine, appendectomy, splenectomy, and right ovarian cystectomy. Enteric lesions were removed through a gastrotomy, 11 enterotomies, and a sigmoid colotomy. Palpation of a lesion on the posterior gastric corpus revealed the absence of transmural involvement, so the lesion was resected above the proper muscular layer. Palpable lesions on the small intestine  level was 7–8 mg/dL. GI endoscopy, including transanal double-balloon endoscopy and capsule endoscopy, and contrast-enhanced computed tomography (CT) revealed multiple VMs on almost the entire GI tract, including the stomach, duodenum, jejunum, ileum, and colon Figures  and 3. Vntestine  were marntestine . For trantestine . The numBRBNS is a rare disease characterized by multiple VMs in the skin and the GI tract , 2. The α, and β-blocker has been attempted [Treatment of BRBNS is directed to the symptom. Gastrointestinal lesions may manifest clinically with chronic continual bleeding. Conservative treatment by oral iron supplementation or blood transfusion is usually sufficient. Pharmacologic treatment to control GI bleeding with agents such as corticosteroids, interferon-ttempted , 13, 14,ttempted , 16. Surttempted . Combinettempted . We perfThere are 91 cases of BRBNS with GI VMs reported in the literature identified from MEDLINE . HoweverIn conclusion, we report a case of BRBNS that required a second surgery for delayed recurrence. Long-term follow-up is important in the BRBNS patients following even successful resections.


Isolated small bowel perforation is a rare presentation of blunt abdominal trauma, and most cases present immediately following the trauma. Delayed presentation of such cases beyond one week of trauma is extremely rare, and various pathophysiological mechanisms were described for the same. We present a 20-year-old male patient who sustained blunt abdominal and pelvic trauma, underwent open reduction and internal fixation for right acetabular fracture, and later developed features of acute peritonitis after one month. On laparotomy, complete terminal ileal transection was found and an ileostomy was done. Delayed perforation of the intestine following trauma occurs due to ischemic necrosis, either through direct trauma to the intestinal wall or indirectly by injury to the mesenteric vessels. Direct trauma to the bowel can result in large hematomas on the bowel wall, which can later perforate due to ischemia. Surgeons should be aware of this rare presentation as the management is challenging and it poses significant medico-legal sequel. Close monitoring of the patient’s vitals and examination for the development of abdominal signs along with repeat imaging at the onset of abdominal signs are cornerstones for successful management of these patients. Blunt abdominal trauma is a challenging condition causing significant morbidity and mortality, especially in developing countries. The delayed onset of symptoms and signs in these patients contributes greatly to the increased morbidity and mortality in these patients. We present a rare case of delayed presentation of small bowel injury following blunt abdominal trauma and has outlined the pathogenesis and management of such presentations.A 20-year-old male met with a road traffic accident when he was hit by a moving car while riding a two wheeler. He sustained blunt trauma to his abdomen and pelvis. After initial resuscitation at an outside hospital, he was found to have right acetabular fracture for which he underwent open reduction and internal fixation. His post-operative period was uneventful. One month after the trauma, he developed abdominal pain and abdominal distension for one day. He also complained of multiple vomiting episodes for the same period. On examination, the patient was conscious, oriented and was afebrile. His pulse rate was 100/min and blood pressure was 110/70 mm Hg. His abdomen was uniformly distended with diffuse tenderness and guarding. Bowel sounds were absent. His chest X-ray revealed free air under diaphragm and X-ray pelvis showed status internal fixation of right acetabulum with plates and screws  scan with contrast can aid in diagnosis. An X-ray can show air under the diaphragm in case of bowel perforation. Focused assessment with sonography in trauma (FAST) is a rapid and sensitive method for detection of free intraperitoneal fluid, but it is less accurate in detecting mesenteric ischemia. Findings such as portomesenteric vein gas and intramural gas in the small bowel in the CT scan are suggestive of severe mesenteric ischemia. A contrast CT scan is the imaging modality of choice in stable blunt abdominal trauma patients who continue to have abdominal pain, in spite of its significant false-negative rate . A repeaLaparoscopy can be used as a diagnostic as well as a therapeutic modality in these cases, and they are shown to prevent non-therapeutic laparotomy in up to 40% of the cases . DiagnosStable patients without clinical signs of peritonitis are managed conservatively. However, the potential for delayed small bowel perforation should be borne in mind and patients warned about the symptoms and counseled regarding the same. Unstable patients and those with evidence of bowel perforation should undergo a laparotomy. Intra-abdominal contamination is usually present and a definitive repair or an anastomosis is likely to break down. There is a threefold increase in morbidity and mortality due to the delayed presentation. There is an increased incidence of postoperative complications like wound infection, wound dehiscence, intra-abdominal abscess, acute respiratory distress syndrome and sepsis adding to the morbidity .The rarity of isolated small bowel perforation, the paucity of clinical findings on initial admission and limitations of current diagnostic investigations lead to a delay in the definitive treatment of these patients. A detailed history including the mechanism of injury, close observation with repeated physical examinations, thorough investigations with CT and/or FAST, repeated if necessary, and awareness of the possibility of small bowel perforation are cornerstones to successful management in patients with blunt trauma of abdomen.


Osteosarcomas of the head and neck region are rare entities that comprise < 10% of all osteosarcomas. Multimodality treatment of patients with osteosarcoma is well-established for osteosarcoma in long bones, and the benefits of chemotherapy in long bones are clearly known. However, there is no consensus regarding the effects of chemotherapy in cases of head and neck osteosarcoma. The prognostic factor for head and neck osteosarcoma is complete tumor resection with negative margin, which is a radical surgery. However, a clear margin may be difficult to achieve in the head and neck region.We present a case of a 69-year-old Japanese woman who developed osteosarcoma of the condyle within the masticator space and infratemporal fossa, which was treated with radical surgery using a modified preauricular and transmandibular approach. Although we recommended adjuvant treatment after surgery, the patient refused this treatment. There was no evidence of local recurrence or distant metastasis through 30 months of follow-up.Our modified preauricular and transmandibular approach allowed access to the masticator space and infratemporal fossa, thereby increasing complete resection of the tumor and resulting in minimal functional and cosmetic deficits. Osteosarcomas at all sites comprise approximately 40–60% of primary malignant bone tumors . OsteosaIn our patient, we performed surgical management via a modified preauricular and transmandibular approach for osteosarcoma of the mandibular condyle that reached the masticator space and infratemporal fossa. This method makes it easy to approach from the glenoid fossa to the infratemporal fossa with a wide field of view by adding a temporal incision and tracing the facial nerve to the conventional preauricular and transmandibular approach.A 69-year-old Japanese woman was referred to our department in August 2015, complaining of swelling in the right preauricular region. She had shown a mass lesion of the right lung on a chest x-ray and was referred to the university hospital. Fluorodeoxyglucose-positron emission tomography (FDG-PET) examination showed accumulation indicative of a pulmonary lesion and a temporomandibular joint lesion. The temporomandibular joint lesion had been enlarging but was otherwise asymptomatic. Her medical history included treatment for pulmonary tuberculosis at the age of 13 and surgery to remove breast cancer at the age of 53.On examination, a tender mass in the right preauricular region was palpable. The chin of the mandible was deviated to the right side during mouth opening . Facial nerve function and mandibular nerve were intact. There was no indication of cervical lymphadenopathy.A panoramic radiographic examination showed resorption of the right mandibular condyle to the ramus Fig. . ComputeA malignant tumor was suspected after analysis by various modalities. Therefore, we performed an incisional biopsy via preauricular incision. Histopathologically, the tumor was largely composed of proliferative, atypical, spindle-shaped cells. Some tumor cells showed increasing mitotic change and extreme atypia Fig. . The hisAfter preauricular temporomandibular incision, the superficial temporal fascia and temporal fascia were elevated. The facial nerve  was protected by the fascia Fig. a.Fig. 5SThe facial nerve trunk was identified, and its branch was traced according to the conventional method.A midline lip-splitting incision was connected to the submandibular incision and accessed the anterior mandibular ramus. The lip-split incision traversed to the periosteum of the mandible; the periosteum dissector was used to elevate the soft tissues of the mandible, and the lateral mandible was exposed. The branch of the facial nerve was traced continuously with the skin flap, and the masseter muscle and deep parotid gland were dissected on the tumor side Fig. c. The maThe surgical defect was reconstructed with a free vascularized fibula with skin paddle Fig. e. The ocThe resected specimen exhibited nearly identical histological findings as observed in the biopsy. Because it involved a periosteal reaction corresponding partially to Codman’s triangle, the tumor was thought to have derived from bone Fig. . ImmunohThere has been no evidence of local recurrence or distant metastasis through 30 months of follow-up. The chin of the mandible deviates to the right side during opening . Centric occlusion has not changed. Facial nerve dysfunction gradually improved and became mild according to House-Brackmann Scale evaluation Figs.  and 8.FiThe most important prognostic factor for osteosarcoma in the head and neck is complete tumor resection with negative margin in radical surgery –10. HoweRegarding surgical margins, there have been reports suggesting the establishment of a margin of 2 cm of bone after effective preoperative chemotherapy ; the safen bloc resection of the tumor and preservation of the facial nerve without cervical branching. There have been various reports regarding approaches to tumors in the infratemporal fossa [In our patient, because the tumor was located in the masticator space and infratemporal fossa and had originated from the mandibular condyle, a modified preauricular and transmandibular approach was selected for complete al fossa –16. ThisExcision using this approach can entirely remove the tumor, including the medial and lateral pterygoid muscle, mandibular ramus, posterior of maxilla, and pterygoid process, with the skull base as the upper border of the surgical margin. Sacrificing the mandibular ramus above the angle of the mandibular condyle allows a wide approach to the glenoid fossa and lower portion of the infratemporal fossa. In addition, the temporal muscle can be dissected from the lateral side and exposed to the infratemporal fossa under direct visualization. Then, the foramen ovale and foramen spinosum can be exposed, and dissection of the mandibular nerve (V3) and treatment of the oval plexus and middle meningeal artery can be performed. It is advantageous for hemostatic treatment to be performed under direct view.The facial nerve and its branches  may preserved.Exposure of the glenoid fossa, pterygopalatine fossa, and infratemporal fossa can be readily obtained.The mandibular division of the trigeminal nerve and the pterygoid plates can be exposed.Flaps for reconstruction are available within the surgical field.This approach has the following advantages:This approach can also be applied to the resection of maxillary cancer that has developed backward, such as within the pterygoid muscle and process.Disadvantages include that the result is not good aesthetically, because there is an incision in the median of the lower lip; however, by extending the incision of the temporal and submandibular regions, this lower lip incision may be avoided.This method makes it easy to approach from the glenoid fossa to infratemporal fossa with a wide field of view by adding a temporal incision and tracing the facial nerve to the conventional preauricular and transmandibular approach.This approach is a suitable method for cases requiring complicated excision of temporomandibular joint, masticatory muscles, and pterygoid process in tumors extending from the glenoid fossa to the infratemporal fossa, and it can be suppressed to minimal functional and cosmetic deficits, including preservation of the facial nerve.A clear margin may be technically difficult to achieve in the head and neck region, particularly in the masticator space and/or infratemporal fossa, because the surgical field of vision is poor, and it is difficult to control bleeding. This modified preauricular and transmandibular approach allows access to the masticator space and infratemporal fossa, as shown by anatomical dissection, thereby increasing the complete resection of the tumor and resulting in minimal functional and cosmetic deficits.


Herlyn-Werner-Wunderlich syndrome is a very rare congenital genitourinary anomaly characterized by uterus didelphys, blind hemivagina and ipsilateral renal agenesis.Authors present a case of Herlyn-Werner-Wunderlich syndrome in a 19-year-old unmarried woman who presented with pelvic pain and pelvic mass. MR imaging revealed the typical features of didelphys uterus, obstructed right hemivagina and ipsilateral renal agenesis. The patient subsequently underwent surgery.Herlyn-Werner-Wunderlich syndrome would be suspected in patients with unilateral absent kidney and pelvic mass. Ultrasonography and MR imaging can well depict the disease entity and surgery is the treatment of choice for obstructed hemivagina. Herlyn-Werner-Wunderlich syndrome (HWWS) is name of a congenital anomaly characterized by uterus didelphys, blind hemivagina and ipsilateral renal agenesis  thereforA 19-year-old, unmarried woman presented with complaint of chronic pelvic pain and a palpable mass at the lower pelvic midline region. On physical examination, a mobile and painless mass was palpated at the lower pelvic midline region. The external genitalia were normal in appearance. Transabdominal ultrasonography reported a cystic structure in the lower pelvic region communicating with the uterus (likely dilated vagina) with endometrial cavities and absent right kidney. A cystic structure with internal septae was also reported in the left adnexal region.MR imaging with contrast was performed and showed duplication of the uterine bodies, endometrial canals, uterine cervices and vaginal canals. The right vaginal canal was significantly dilated. There was communication between the two cervices well seen in the axial T2W sequence. A small tubular structure with internal fluid signal along the anterolateral aspect of the dilated right hemivagina represented blind ectopic ureter (the mesonephric remnants) Figs.  and 2. RA heterogeneous cystic structure was seen in the left ovary with hemorrhagic components.The patient underwent surgery under general anesthesia. The septum was excised and hematocolpos was drained. The hemorrhagic, infected left adnexal cyst was also excised.HWWS is a very rare congenital anomaly . It reprLan Zhu et Al. recommend that the syndrome be classified to two types (classification 1 and 2) according to the complete or partial obstruction of the hemivagina as the clinical details associated with each type are distinctly different [In classification 1.1, the affected hemivagina is completely obstructed and the uterus behind the septum is totally isolated from the contralateral one. In classification 1.2, the hemivagina is completely obstructed; the cervix behind the septum is maldeveloped/atretic and menses from the uterus behind the septum cannot drain through the atretic cervix. In classification 2.1, there is small communication between the two hemivaginas, which makes the vaginal cavity behind the septum incompletely obstructed. In classification 2.2 the hemivagina is completely obstructed, and a small communication exists between the duplicated cervices (as is in current case). Menses from the uterus behind the septum can outflow through the communication to the offside contralateral cervix. Because the communication is small, the drainage is still impeded [The clinical presentation mainly is related to the type of the abnormality. In classification I, the common clinical presentation can be pelvic pain shortly after menarche, associated with vaginal or pelvic mass .In classification 2, the clinical presentation may be delayed (as in current case), as the obstructed side can be drained through the contralateral vagina. Acute onset of abdominal pain, fever and vomiting can be the presenting symptoms due to bleeding from the fallopian tube . EndometThe mainstay of imaging work up is ultrasonography and Magnetic Resonance (MR) imaging. Ultrasonography can show uterovaginal duplication, hematocolpos or hematometrocolpos along with the absence of ipsilateral kidney , while MIn patients with Classification 2.2, hysterosalpingography can also show that contrast material passes through the communication between the duplicated cervices to the contralateral uterus and then the cavity behind the septum .Treatment of choice for obstructed hemivagina is resection of the vaginal septum  which caLan Zhu et Al. performed a retrospective long-term follow-up study on surgical prognosis and pregnancy outcomes and found that full resection of the vaginal septum was associated with good outcomes and fertility. No pathologic pregnancies or pregnancy complications were documented . GholoumAs the conclusion; HWWS is a rare clinical entity, however it can be suspected in female patients with unilateral absent kidney and pelvic mass. Ultrasonography and MR imaging can well depict the disease entity and surgery is the treatment of choice for obstructed hemivagina. Good long-term outcome is expected after vaginal septectomy.


Pseudoangiomatous stromal hyperplasia (PASH) is a benign breast condition of collagen proliferation. In this article, four cases are presented in series to illustrate the varying clinical presentations of PASH at mammography and sonography, as well as the vastly differing age groups that can be affected. A literature review of the aetiology, pathology and management of PASH is included to provide a comprehensive but succinct overview of the condition, what it is and how to recognise and manage it. The cases highlight the existence of PASH, its varied forms of presentation and the implications for further patient management.Although uncommon, it is important to be aware of and recognise this condition in order to avoid submitting the patient to unnecessary surgery or follow-up examinations once it has been diagnosed.Miss J, a 40-year-old asymptomatic woman, presented for routine screening. Her mammogram  revealedMrs K, a 50-year-old asymptomatic woman, was found to have probably benign nodules in her right breast at previous sonography 12 months earlier. At her annual follow-up mammogram and breast sonar, one of these lesions, located superiorly in the right breast, appeared to have increased very slightly in size and become less well defined on sonar . Her mamMiss M, a 14-year-old girl, presented for sonar assessment of a large, growing, palpable lump in her left breast. Corresponding to the lump, there was a 7 cm × 4 cm × 5.7 cm, slightly mixed, predominantly isoechoic, probably benign mass on sonar . BecauseImmunohistochemical stains were as follows:Estrogen receptor (ER) positive in the duct component and negative in the stromal component.Progesterone receptor (PR) positive in the epithelial component and negative in the stromal component.MNR116 positive in the epithelial component and negative in the stromal component.Androgen receptor focal positive in the stromal component.CD34 positive in the vascular element only.Mrs Z, a 71-year-old diabetic woman, presented complaining of a palpable lump in the right breast. Her mammogram showed extremely dense tissue with a grade D pattern and increased density in both upper, outer quadrants . Sonar dIn all cases, there was radiological and pathological concordance. Annual surveillance was advised for cases 1, 2 and 4. Case 3 required no radiological follow-up but was referred for surgical opinion to discuss the option of surgery in view of possible aesthetic problems because of the size and enlargement of the mass and the patient’s preference. However, the patient did not attend her surgical appointment.3 PASH has been shown to occur across a wide age range and has been documented in women from 14 to 67 years old, the majority occurring between 30 and 50 years.4 It is rare over the age of 50, which further correlates with a hormonal aetiology.6 PASH can also occur in men, associated with gynaecomastia.6Pseudoangiomatous stromal hyperplasia is a benign breast condition of collagen proliferation. The exact aetiology and pathogenesis are unknown, but hormonal factors are known to play a role in the development of PASH; it is more common in pre- and perimenopausal women.1 In addition, PASH cases have shown strong stromal cell PR positivity and faint stromal nuclear reactivity for oestrogen was shown in one case, whereas the stromal cell nuclei of control cases without PASH did not stain for either receptor.6In known cases of PASH, the lesion size was shown to change with menses, consistent with a hormone-related fluctuation.4 It may also present as an incidental, microscopic finding7 and may or may not be palpable.2Clinically, PASH usually presents as a mass, typically enlarging, sometimes rapidly.5 On mammography, it typically has the appearance of a probably benign mass,1 lacking calcification within it.8 It varies in size, with measurements of 1 cm–12 cm reported.8 On sonar, it is usually a hypoechoic, oval or round, benign-appearing mass but may be slightly heterogeneous.9 If it occurs within fibroadenosis, the imaging findings reflect this, with mammographically dense tissue and sonographic shadowing, hypoechoic tissue.9Radiologically, the classic description of PASH is of a single, well-circumscribed, round or oval, mobile mass, resembling a fibroadenoma on mammogram and sonar.2On MRI, the findings of PASH are non-specific and range from an enhancing mass to clumped, non-mass-like enhancement, usually with benign kinetics.9 It can thus be associated with any other specific pathology or tissue type and the morphology of this may be the dominant finding.However, as illustrated by the above cases, PASH may present with various other findings, none specific to the condition, and indeed, it is most commonly found as an incidental finding in a biopsy specimen, the biopsy having been done for another reason.6 This is, however, not true angiomatous proliferation and these channels are not blood vessels. Because of this, however, it may be histologically mistaken for a vascular neoplasm.5Breast tissue containing PASH exhibits stromal cell proliferation, that is, proliferation of collagen, with slit-like channels lined with myofibroblasts (spindle cells) resembling vascular channels.α-smooth muscle actin but negative for CD31 and factor VIII (an endothelium-specific marker). In addition, the cells are hormonally sensitive and frequently express PR and less frequently ER.10The spindle cells are positive for Vimentin, CD34, BCL2, CD99 and 11 Within an area of fibroadenosis, the condition may be diagnosed as fibroadenosis. PASH may also be misdiagnosed as a different condition at biopsy when it is present as an incidental finding. In addition, PASH may be confused with other diffuse breast diseases such as diabetic mastopathy.12Radiological differential diagnosis depends on mode of presentation. As a benign-appearing mass, the main differentials are fibroadenoma and phyllodes.The main pathological differential diagnoses are low-grade angiosarcoma and spindle cell containing entities such as phyllodes and desmoid tumours.6 It is not associated with synchronous carcinoma6 .Pseudoangiomatous stromal hyperplasia is a benign condition. It is neither premalignant nor a risk factor for the development of carcinoma.6If the imaging findings are equivocal, a histological examination is mandatory for a definitive diagnosis.A diagnosis of incidental PASH requires no active intervention or radiological follow-up. Pseudoangiomatous stromal hyperplasia is a BIRADS II condition.12 and in patients with discordance following triple assessment.6 After surgery, it has been shown to have a low rate of recurrence (15% – 22%).11Surgery may be performed for enlarging lesions, diffuse PASH with massive breast enlargement which is very rare10 and would be inappropriate in young premenopausal women.There is no established conservative treatment for PASH. It responds to tamoxifen; however, the effects may only be sustained with prolonged therapy. Long-term tamoxifen may not be ideal because of its side effectsInformed consent was obtained from all the patients for inclusion of their images and information in the study.


Solitary fibrous tumor (SFT) derived from the abdominal wall is rare. We report a case of SFT in the abdominal wall. When abdominal computed tomography reveals soft‐tissue mass and analysis of preoperative needle biopsy reveals spindle cell tumor, the possibility of SFT should be considered. Klemperer and Rabin2A 26‐year‐old man presented with a rapidly growing mass in the left abdominal wall Figure . The masIn immunohistochemical staining, the tumor was positive for CD34, B‐cell lymphoma 2 (BCL‐2), and CD99 antigens Figure . Stains 3Solitary fibrous tumor is a rare disease, mostly described in case reports. It is difficult to diagnose and often misdiagnosed as other kinds of spindle cell tumors, especially synovial sarcoma.Most often, SFT is a slow‐growing mass with average diameter of about 10 cm.Preoperative needle biopsy was of limited value in our patient as it did not distinguish between SFT and other spindle cell tumors. Complete surgical resection is the main treatment for SFT4When abdominal computed tomography reveals soft‐tissue mass in the abdominal wall and analysis of preoperative needle biopsy reveals spindle cell tumor, the possibility of SFT should be considered.None declared.Wang S: performed the operations, reviewed the literature, and drafted the manuscript. Guan J: collected the data and helped to draft the manuscript. Liu M: performed the operations. Li S: collected the data. Jiang C: conceived the study. All authors read and approved the final manuscript.


He was initially diagnosed with neurosarcoidosis based on imaging. MRI demonstrated intraparenchymal lesions involving the right temporal lobe and cerebellum. These appeared as extremely hyperintense signals on T2 weighted imaging and as homogeneous enhancements with a small non-enhancing cystic component on contrast-enhanced MR. Diffuse post-contrast enhancement of the craniospinal meninges was also noted. Post-radiation therapy PET-CT demonstrated a highly FDG-avid tumour in the right temporal lobe and left cerebellum. To our knowledge, a multifocal form of protoplasmic astrocytoma in an adult patient has not been previously described.Protoplasmic astrocytomas are a poorly characterized and extremely rare subtype of astrocytoma. We describe the CT, MR and  They are classified by the World Health Organization (WHO) according to histological grade. Low-grade astrocytomas (WHO grade II) are diffusely infiltrating tumours, and can be further subdivided into fibrillary, gemistocytic and protoplasmic subtypes.In aggregate, all three forms of diffuse astrocytomas account for roughly 5% of primary CNS tumours and 10–15% of astrocytic gliomas, with protoplasmic being one of the rarest forms.18F-fludeoxyglucose positron emission tomography (FDG-PET) imaging characteristics of this extremely rare presentation. The multifocal nature of this patient’s low-grade astrocytoma—including involvement of the temporal lobe, posterior fossa and craniospinal meninges—makes this case both unique and significant for the interpretation of the radiological findings associated with adult CNS tumours.Although glioblastomas (WHO grade IV) have been known to have multiple foci, an adult patient with multifocal grade II disease involving multiple lesions in both the brain and the spine has not yet been described. We report such a case, with an emphasis on the CT, MR and A 29-year-old male presented with progressively worsening symptoms, including altered mental status, lower extremity weakness, nausea and vomiting for 6 months, with symptomatology extending back more than 6 years prior to this decline. He also reported headaches, dizziness, multiple falls, and right facial numbness and tingling during his 6-month decline. Previously, he had had seizures that were controlled with medication. Prior to referral to neurosurgery, his presumptive diagnosis was hydrocephalus, for which he had a ventriculoperitoneal shunt placed. Neurological examination was significant for mild dysarthria, a non-reactive left pupil, nystagmus on primary gaze of the left eye, bilateral nystagmus on horizontal gaze and mild action tremors in both hands.T1 weighted MRI of the brain demonstrated extensive post-contrast nodular enhancement of the basimeninges associated with an approximately 5 × 4 cm solid and cystic mass in the right temporal lobe  may have foci with protoplasmic features; thus, it is important to demonstrate that the entire tumour has these features before making a diagnosis of protoplasmic astrocytoma. In the present case, we had only one small  biopsy confirming the diagnosis, and for this reason, the diagnosis of "astrocytoma with protoplasmic features" was made instead of "protoplasmic astrocytoma". Although no biopsy was obtained from the spinal lesions, it was determined that these lesions were of similar aetiology to the biopsy specimen taken from the brain.9 The duration of symptoms ranged from 7 months to 28 years, and all patients presented with seizures. None of the tumours in this study was multifocal in nature and many underwent complete resection. Of the nine patients who were managed surgically, only one had a recurrence.9A 1995 study of 16 purely protoplasmic astrocytomas more frequently observed the tumours in males at a mean age of 20.7 years.10 reviewed the MR images and histopathology of eight consecutive cases of biopsy-proven protoplasmic astrocytomas. They concluded that a primary cerebral neoplasm representing a protoplasmic astrocytoma should be considered as a differential for large frontal or temporal tumours with increased signal on T2 weighted imaging and a large proportion of the tumour demonstrating substantial T2 FLAIR suppression. In our patient, the intraparenchymal brain lesions demonstrated similar findings on T2 weighted and FLAIR MR. However, we observed avid enhancement of the solid components of these lesions in contrast to the findings reported by Tay et al10 that demonstrated none-to-moderate post-contrast enhancement. Furthermore, the intraparenchymal cranial lesions in our case had a large solid component, whereas prior studies included, at most, a minimal nodular component. The leptomeningeal involvement in our patient showed significant post-contrast enhancement and nodular areas of high T2 weighted signal intensity involving the craniospinal meninges and the cauda equina, which is also novel.In a recent series, Tay et alFDG-PET imaging of protoplasmic tumours has not been previously reported in literature. In our patient, the intraparenchymal cranial lesions demonstrated avid FDG uptake, which may correlate with the somewhat aggressive nature of the neoplasm observed histologically.6 This is further complicated by the fact that sarcoidosis presents with neurological involvement at initial presentation in 5–10% of the individuals.4Low-grade astrocytomas are slow-growing and well-differentiated tumours, although there is a tendency for malignant progression that is often refractory to surgical treatment, chemotherapy or radiation therapy. Protoplasmic astrocytomas are a rare variant of diffuse astrocytomas with a generally favourable prognosis. Radiologically, these tumours can mimic neurosarcoidosis, Wegener’s, tuberculosis, leukaemia, lymphoma, primary CNS neuromatosis, metastases and, less commonly, infections, among other rare entities.Our patient’s presentation and clinical course demonstrates the importance of obtaining biopsies in cases of diffuse meningeal enhancement on MRI when a diagnosis is unknown and clinical history supports multiple differential diagnoses. Radiologists and neurosurgeons need to be aware of protoplasmic astrocytomas, as the non-specific radiological and clinical presentations can make this a complex and challenging diagnosis.Protoplasmic astrocytomas are a poorly characterized and extremely rare subtype of astrocytoma. Multifocal variants of this tumour have seldom been reported.Radiologically, these tumours can mimic neurosarcoidosis, Wegener’s, tuberculosis, leukaemia, lymphoma, primary CNS neuromatosis, metastases and, less commonly, infections, among other rare entities.The non-specific radiological presentation makes obtaining a diagnosis difficult, highlighting the need for obtaining a biopsy in cases of diffuse meningeal enhancement on MRI when a diagnosis is unknown.


Sacrococcygealteratoma is very rare in adults with only few cases reported in literature. It is a slow growing benign cystic tumor with 1–2% chance for malignant transformation and may attain a huge size causing pressure effect on pelvic and intra-abdominal organs. When grows caudally, it causes discomfort, skin excoriation and cosmetic disfigurement.We present a giant, long standing Sacrococcygeal teratoma in an adult female patient which caused her significant obstructing lower gastrointestinal and urinary symptoms besides the discomfort and disfigurement of the sacral region.Sacrococcygeal teratomas are commonly present early in life but are very rare to be seen in adults with such a huge size in such a place where it interfered with patient sitting and sleeping.Diagnosis of our case was suspected by clinical examination, ultrasound and MRI while histopathology confirmed the diagnosis. It was completely excised along with the coccyx with primary wound closure and had a good postoperative recovery. Up till now, 18 months follow-up using clinical, biochemical and radiological assessment revealed no evidence of recurrence.Sacococcygeal teratoma should be considered in the differential diagnosis of large pelvic masses in adults. In the presence of an extra-corporal and deep intra pelvic extension, we recommend the posterior perineal approach for excision as it has convenient control over the mass during surgery and good cosmetic results. These tCysts may be filled with serous fluid, mucoid, or sebaceous material and lined by true epithelium.Altman classified SCTs into 4 types according to its position: type I, predominantly external mass with a small presacral component; type II, external mass with a intrapelvic component; type III, external mass with a pelvic and abdominal component; and type IV, internal mass with an intrapelvic and abdominal location; types II and III are usually dumb-bell shaped [The clinical presentations of the teratoma are mainly due to its mass effect on the adjacent organs as the rectum, urinary bladder and pelvic vasculature causing obstructive symptoms of the affected system. Projection of the mass externally will cause disfigurement and discomfort in addition to painful skin excoriation and ulcers.CT scan and or MRI imaging are invaluable in the diagnosis of these lesions and in delineating their origin, extent and relation to the pelvic and abdominal organs. Surgical excision is considered curative when the coccyx is removed with the mass and the histo-pathological report confirmed the benign nature of the teratoma.Our work has been reported in line with the SCARE criteria .2A previously well 49- year old multiparous Jordanian female who was not on any medication referred to our teaching hospital complaining of the presence of a huge sacrococcygeal mass of eight years duration. The mass had progressively increased in size from a small painless nodule at the tip of the coccyx till it became large enough to cause obvious cosmetic disfigurement of the body contour and shape. It prevented the patient from lying on her back or sitting down comfortably. For the last year, patient started to complain of increased urinary frequency and constipation with a narrow caliber fecal stream. Few weeks prior to her visit, she started feeling progressive deep abdominal and pelvic pain and noticed engorgement of her lower limbs which compelled her to seek medical help.Inspection showed a huge smooth rounded mass (20 × 20 cm) extending down the lower end of the natal cleft hanging over buttocks as shown in Digital rectal examination revealed a tense smooth slightly tender bulge encroaching on the posterior wall of the rectum with a normal feeling mucosa.Magnetic resonance imaging studies (MRI) showed a well-defined encapsulated mass of 25, 15, 10 cm size. The mass was filling the presacral space, extending up the pelvis and down posterior to the rectum and anal canal compressing both and the urinary bladder with a huge extra corporal extension It appeared hypo-intense in T1 weighted images with faint calcification spots while it showed hyper intensity in T2 imaging with clear surrounding boundaries except for a close relation to the coccyx. There was no enhancement of the lesion and no invasion of the adjacent structures nor was bone erosions. MRI findings were interpreted as a Sacrococcygealteratoma, possibly originating from the coccyx. However, the differential diagnosis was going around other rare conditions as rectal duplication cysts, lipoma, liposarcoma, sacrococcygealchondromas and chordomas.Related serum tumor markers as alpha-feto protein, human chorionic gonadotropins (HCG), carcinoembryonic Antigen (CEA), Lactate Dehydrogenase (LDH) Carcinoma Antigen (CA 125) were all normal .Fig. 3CyFine needle aspiration was tried twice but did not yield any fluid or cells for cytological examination.Under general anesthesia, the patient was placed in prone position and an elliptical incision was performed as in posterior perineal approach. The mass was dissected and followed up into the pelvis where it was separated from the pelvic viscera easily but when followed downwards it was found firmly attached to the coccyx. Resection of the coccyx was performed to complete the excision of the mass and the wound was closed in the midline after securing hemostasis.The patient had smooth recovery with nice wound healing and cosmetic appearance . The finPatient was seen regularly for 18 months by now, and a recent MRI showed no recurrence.3Teratomas are neoplasms composed of mixed tissue elements derived from two or more germ cell layers. While the majority of teratomas present congenitally in the sacrococcygeal region, they have been identified throughout the body .Sacrococcygealteratoma is one of the commonest fetal neoplasms, but is so rare in adults that only case reports and few case series were reported on reviewing the literature with lack of information of its incidence in the general population ,2.Both infantile and adults types of sacrococcygealteratoma tend to occur more frequently in females with a female-to-male ratio of about 4–10:1 .Adult SCT is mostly seen as an intra-pelvic mass when compared to the neonatal variety which mostly presents as externally visible mass . TeratomIn comparison to the infantile type, the incidence of malignant transformation of the adult teratomas is much lower with an incidence of about 1–2% .As SCT mostly grows slowly within the pelvic cavity it may cause no symptoms and discovered during investigation for another intra- abdominal pathology. However, by increasing in size, it may present as deep pain and discomfort because of the mass stretching and compression effects. Larger mass could also cause symptoms secondary to its pressure effect on the pelvic viscera. Huge teratomas may extend externally to cause cosmetic and body contour disfigurement as was also noticed in our case .Ultrasonic examination of the cyst usually reveals heterogenic echogenicity with cystic and solid areas ,6. CompuBlood investigations might be helpful in suggesting the possibility of malignancy. This could be seen when the tumor markers as alpha fetoprotein, carcino-embryonic antigen and human gonadotrophin are elevated ,8. Our pPre-operative pathological studies like trans- cutaneous or trans-rectal biopsy may not be advisable because of the risk of recurrence or dissemination in case of malignancy ,9. Fine Exploration, dissecting the mass from the surrounding viscera and excising the attached coccyx is the modality of choice to remove the cyst and its midline origin ,11.In our case, the histopathological examination of the specimen proved the benign nature of the cyst as a mature teratoma.If histopathological examination showed malignant differentiation then; the possibility of recurrence could be avoided by postoperative chemotherapy and radiotherapy .The operative excision is usually accomplished through laparotomy or laparoscopic exploration. However, because of the significant extrapelvic and perinealextention, we have chosen the perineal approach which proved to be helpful and ended with a good cosmetic result. This, we think, makes our case unique among other reported cases.After repeated postoperative follow-up visits to the clinic; the patient confirmed the return of her urinary and defecation habits to normal and was happy with the satisfactory result of the healed wound.4Although SCT is a rare tumor in the adults, still it should be considered in the differential diagnosis of a pelvic mass especially when associated with extra-pelvis extension. Surgical excision of the tumor with coccygectomy is the treatment of choice.Posterior perineal approach is recommended in case of extra-pelvic extension as it gives good cosmetic look in addition to convenient control over the mass during surgery.We declare that we have no competing interests.This research received no specific grant from any funding agency in the public, commercial, or non-for-profit sectors.Institution Review Board (IRB) of the Jordan University of Science and Technology and King Abdulla University Hospital granted the approval for all the work done in these institutions.Written informed consent was obtained from the patient for publication of this case report and accompanied images.1. Nawaf J Shatnawi: Took part in the surgical management, participated in the design and Coordination of the study helped to draft the manuscript and reviewed the literature.2. Muhammad R Khammash: Participated in the interpretations of the radiological images, helped to draft and editing the manuscript.3. Abddel karim H Omari: Took part in the surgical management, Participated in the design, studied the images and reviewed the literature.All three authors read and approved the final manuscript.Not applicable.Nawaf J. Shatnawi.Not commissioned externally peer reviewed.


We report an uncommon case of posterior epidural migration of a lumbar disc fragment (PEMLDF) in a patient presenting with acute, progressive back pain, radiculopathy, and weakness. PEMLDF can be mistaken for neoplastic or infectious etiologies on imaging, presenting a diagnostic and management challenge. Our patient underwent an urgent decompressive lumbar laminectomy, which revealed a PEMLDF intraoperatively. He went on to achieve good neurologic recovery. A herniated lumbar disc is characterized by the displacement of disc material beyond the normal confines of the fibrous annular ring. Due to the presence of the posterior longitudinal ligament, these fragments most often travel in a posterolateral direction . PosteriWe present a 40-year-old male with a history of chronic back pain and a recent, acute progression of bilateral lower extremity paresthesias and weakness. Over a seven-day period prior to presentation, the patient experienced a sudden onset of bilateral leg numbness, with no history of trauma or another precipitating event. The right leg was affected more severely than the left, traveling primarily in a distribution down the lateral leg into the dorsum of the foot and great toe. He eventually began having a shooting pain in this same distribution. He also reported a progressive weakness with bilateral ankle and toe dorsiflexion and five days duration of perineal numbness and mild urinary retention. On exam, he was unable to dorsiflex his ankles or great toes against gravity and had reduced sensation in the lateral aspect of both lower legs and the dorsum of bilateral feet. The patient’s Achilles reflexes were also diminished bilaterally, though reflexes were present and brisk at the knee. It is also pertinent that no upper motor neuron signs were present.Magnetic resonance imaging (MRI) of the lumbar spine revealed a somewhat heterogeneous, but predominately T2-weighted, hyperintense mass in the left lateral and dorsal epidural spaces Figure . There wDue to the acute onset of symptoms, as well as the severity of neurologic involvement, the patient was taken to surgery for exploration and removal of the epidural mass. An L4-5 laminectomy was performed, which revealed a very large dorsally migrated disc fragment that erupted as soon as the ligamentum flavum was removed. The large mass, which was displacing the thecal sac to the right, was removed in several large pieces, tracking down to the L4-5 disc space until all neurologic elements were satisfactorily decompressed. At that time, the annular tear was visualized on the left-hand side; the fragment was sent to pathology and confirmed to be disc material. Confident that the extruded disc material was completely removed, the wound was irrigated and closed uneventfully.Postoperatively, his leg pain and saddle anesthesia resolved immediately. By the six-week follow-up appointment, he had regained full strength in his ankles and reported no bowel, bladder, or sexual dysfunction.Lumbar disc herniations are common and typically follow an expected trajectory, most often in a posterolateral direction, and less frequently foraminally or centrally . They wiThe diagnosis of this rare form of herniated disc is confounded by atypical imaging and symptoms mimicking an abscess, hematoma, facet cyst, or neoplastic process, leading to diagnostic difficulty and uncertainty in management. The reported patient presented a diagnostic challenge due to the relatively nonspecific imaging characteristics seen on magnetic resonance imaging (MRI). Although disc fragments typically are isointense on T1-weighted images and hyperintense on T2-weighted imaging, these findings are nonspecific and can overlap with other diagnoses when present in the dorsal epidural space, such as abscesses, hemorrhage, or synovial cysts . If a coTreatment strategies for the components of the differential diagnosis may vary significantly, highlighting the importance of early diagnosis and appropriate clinical suspicion. For example, while a PEMLDF will typically be treated with surgical resection, treatment for an epidural abscess or hematoma may be quite different. Treatment options for an abscess include intravenous antibiotics, percutaneous aspiration, or surgical drainage. Similarly, depending on the neurological status of the patient, an epidural hematoma may require surgery or may be observed. Importantly, however, early diagnosis and treatment of PEMLDF have been shown to correlate with a positive clinical outcome. In a study from 2011, early surgery within three days of symptom onset was one of two main factors contributing to a good or excellent prognosis using the modified Odom criteria . IdentifPEMLDF is rare and provides a diagnostic challenge, sometimes mimicking other, more common posterior epidural etiologies. Characteristics seen on MRI or physical exam can suggest the presence of PEMLDF. This diagnosis, however, requires a high clinical suspicion. Timely diagnosis is of utmost importance, as early surgery has shown to be beneficial for positive clinical outcomes.


Patients with appendicular endometriosis (AE) constitute <1% cases of all pelvic endometriosis cases. AE presents with pain in right iliac fossa and symptomatically mimics appendicitis and definitive diagnosis is possible only after histopathological examination of excised appendix.In this case report, we present a rare case of appendicular endometriosis in a young woman suffering from pain in right iliac fossa and periumbilical region (on/off) for the past one year. She had a past history of infertility. Blood investigations were normal and CT scan of abdomen demonstrated findings suggestive of appendicitis. Laparoscopic appendicectomy was performed, which revealed 2 cm tumor-like mass (1.5 cm diameter) present at the tip of appendix involving the mesoappendix. There was no ascites, peritoneal or omental deposits or any signs of inﬂammation. Post-operative recovery was good. Dissected appendix specimen was sent for histopathological examination which confirmed endometriosis of appendix.Gastrointestinal endometriosis (GE) accounts for 3–37% of all endometriosis cases, whereas AE is present in only ∼3% of all GE cases and constitutes <1% of all the endometriosis cases. AE frequently involves tip and body of the appendix. The layers of appendix most commonly affected are muscular and seromuscular (∼2/3rd cases), followed by the serosa (∼1/3rd cases).Thus, appendicular endometriosis is a rare entity and should be included in differential diagnosis in young women presenting with pain in right iliac fossa and having history of infertility. In majoThe presence of endometrial glands and stroma outside the uterine cavity is known as endometriosis . It rema2A 35-year-old female came to the surgical out-patient department of our hospital with chief complaints of intermittent pain in the periumbilical region and right iliac fossa for one year. Pain was not associated with other complaints or menstruation. There was a history of infertility and no other significant past medical history. The physical examination was unremarkable. The routine blood investigations were normal. USG abdomen and pelvis revealed well-defined nodular lesion (1.5 × 1.3 × 1.6 cm) in right iliac fossa, and mild hypoechoic circumferential wall thickening in the terminal ileum and ileo-caecal junction (3–5 mm thick). Contrast enhanced computed tomography (CT) of abdomen revealed well-defined long tubular structure  with lobulated tip along the postero-medial wall of caecum, mild pericaecal stranding and thickened right lateral conal fascia – suggesting of acute appendicitis . AdditioIntra-operatively, there was presence of a 2 cm tumor-like mass at the tip of appendix involving the mesoappendix, while base of the appendix was normal. There was no ascites, peritoneal or omental deposits or any signs of inﬂammation. The entire appendix measuring 2.5 × 2 × 5 cm was dissected and excised . Caecum 3Endometriosis is described as an estrogen-dependent, chronic inflammatory disorder. Presence of viable endometrial tissue outside the uterus is the characteristic finding. It frequently involves the reproductive organs and peritoneum. It may also affect the GIT, omentum, mesentery and surgical scars, while the distant and rare sites includes the lungs, kidneys, nasal cavity and skin. Gastrointestinal endometriosis (GE) accounts for 3–37% of all endometriosis cases, whereas AE is present in only ∼3% of all GE cases and constitutes <1% of all the endometriosis cases .The most frequent symptoms includes dysmenorrhea followed by chronic non-menstrual lower abdominal pain. Other symptoms are infertility, painful coitus, pain during defecation, painful bladder symptoms and dysuria, bowel symptoms , and ovarian mass . ChronicNone of the available diagnostic procedures  help in the preoperatively diagnosis of AE. A definitive diagnosis is possible only after histopathological examination of the excised appendix. Since laparoscopy provides a direct clue due to visualisation of features under magnification, it may be considered as a standard diagnostic method. However, when a young woman presents with complaints of chronic pelvic non-menstrual pain, and past history of infertility and pelvic endometriosis, diagnosis of AE must be suspected. Similarly, our patient was young and had a history of chronic lower abdominal pain and infertility, which helps in the diagnosis. Also, CT scan revealed findings suggestive of acute appendicitis.AE frequently involves tip and body of the appendix. The layers of appendix most commonly affected are muscular and seromuscular (∼2/3rd cases), followed by the serosa (∼1/3rd cases). American Society for Reproductive Medicine has proposed a staging system, which categorises the endometriosis in to 4 stages: Stage I to IV i.e., minimal, mild, moderate and severe . This stSurgical excision of affected tissue/organ is generally needed for the most efficient and exhaustive treatment of endometriosis. As per the recent guidelines, appendicectomy should be performed laparoscopically, unless contraindicated . The man4Appendicular endometriosis is a rare entity and almost always diagnosed after surgical excision of the appendix followed by histopathological examination. However, it should always be included in differential diagnosis when a young woman presents with complaint of chronic right iliac fossa pain mimicking appendicitis and has a history of infertility.The authors declare that they have no conflict of interest.This study did not receive any funding.The study was approved by Ethics Committee of Hospital Sahloul Sousse.Written informed consent was obtained from the patient.Study concept or design – AS, HA.Data collection – HA, WF, RG.Data interpretation – MBL, WF, RG.Literature review – WF, ABA, SL.Drafting of the paper – HA, MA, SL.Editing of the paper – AB, AS, AM.As this was a case report and not a clinical trial, this study does not require registration.Houssem Ammar.Not commissioned, externally peer-reviewed.


Bleeding in the gastrointestinal tract is a common complication of oral anticoagulant therapy (AT), and it usually appears as mucosal erosion or ulcer; however, intestinal submucosal hematoma (ISH) is an uncommon cause of hemorrhage.This report presents the case of a 70-year-old woman with acute hematochezia induced by AT. She underwent computed tomography and endoscopy.Colon submucosal hematoma.Conservative treatment had no effect, and the patient underwent emergency surgery.Surgical resection showed hemorrhage and necrosis in the left colon, and the patient recovered 24 hours after surgery and continued AT.The present case indicates that the ISH should be kept in mind as a complication of AT. It can be managed conservatively in some stable patients, but emergency surgery may be needed in some serious situations. Further, submucosal hematoma is rarely reported. A review of related literature revealed that the most common sites are the esophagus and duodenum; moreover, small intestinal submucosal hematoma (ISH) has been reported in 1 per 2500 patients on AT each year, and colon submucosal hematomas have been described less frequently in the literature. The clinical symptoms of colonic submucosal hematomas typically include abdominal pain, intestinal tract obstruction, bleeding, and so on. Most ISH cases were diagnosed by endoscopy, and radiological examination, or both.–7 Even in some cases of submucosal tumor, patients required surgical intervention for diagnosis and treatment. We propose that endoscopic ultrasonography (EUS) would help in the differential diagnosis of similar or atypical lesions, as, to our knowledge, this has not been reported. ISH can be treated conservatively, but surgical treatment is necessary in some patients under serious conditions or when conservative management is unsuccessful. Herein, we present a case of hematochezia arising from the colon submucosal hematoma due to AT, but EUS was not used to confirm the diagnosis because it developed rapidly and was diagnosed clearly using endoscopy and computed tomography (CT).Many clinical conditions can lead to lower alimentary tract bleeding, including tumor, ischemic bowel disease, and ulcerative colitis. Recently, with the increased use of anticoagulant therapy (AT), such as warfarin and heparin, the incidence and complications of alimentary tract bleeding have become more common.2A 70-year-old female patient was admitted to our cardiovascular department for chest distress for 11 hours. The patient presented with shortness of breath at rest and difficulty in laying in the recumbent position. She had been diagnosed with hypertension and diabetes mellitus several years prior and was on oral medication for their treatment. After clinical examination, she was diagnosed with coronary atherosclerotic heart disease and subsequently underwent coronary angiography and stent implantation , prolonged prothrombin time , and an international normalized ratio of 1.2 . Colonoscopy revealed a submucosal mass, covered with a partially ruptured mucous membrane and with some clots in the cavity, which caused obstruction in the sigmoid colon, hindering further examination , luminal narrowing, and intestinal obstruction. Moreover, 2 additional signs were commonly seen in intramural hematoma: “coiled spring” and “pseudokidney” signs. Endoscopically, it presents as blue submucosal mass with or without a visible tear, and when the mucous membranes covering the hematoma were peeled off, a shallow ulcer formed over a wide area with scars appearing after about 1 month. If the patient is stable, but with an unclear diagnosis, a pathological finding may help differentiate between a hematoma and a neoplasm, but a colonoscopic biopsy may result in bleeding. A previous study reported that this disease was misdiagnosed as neoplasm or false aneurysmal tumor, and a final diagnosis was obtained by laparotomy. Recently, EUS has emerged as a useful diagnostic tool that facilitates determination of the thickness of the wall from which the lesion has originated. In addition, the technique facilitates the assessment of echostructures of the lesion and the thickness of the infiltration, and it enables the physician to perform thin-needle or core-needle biopsy to collect materials for cytological and histopathological analyses. In the present case, abdominal CT revealed bowel wall thickening and hyperdensity in the luminal area, whereas endoscopy showed that the hematoma ruptured into the bowel cavity with stenosis. Moreover, the pathological examination excluded any possibility of malignancy, so we diagnosed the case as ISH.The prothrombin time and international normalized ratio are usually abnormally prolonged,,7,12–15 Surgical intervention should be reserved for those who exhibit either a deteriorating condition or an unrelenting intestinal obstruction or for those with signs of bowel necrosis or peritonitis. Recently, several new therapeutic strategies, as alternatives to surgical treatment, have been reported, such as percutaneous ultrasonically guided drainage and balloon dilatation using endoscopic incision and drainage. However, they pose a risk for intestinal perforation. Ulla-Rocha et al reported 2 cases of perigastric hematoma and perirectal hematoma, which were treated successfully with endoscopic ultrasound-guided fine needle aspiration (EUS-FNA)-guided drainage and stent placement, and they recommended it as a safe method for chronic hematoma. To our knowledge, ISH treated with EUS-FNA had not been described previously. As “tamponade effect” could lead to further bleeding, the exact duration and time of drainage should be further evaluated. In this case, self-drainage was performed after the mucous membrane ruptured into the intestinal cavity, but as conservative treatment was not successful and there were signs of bowel necrosis, emergency surgery was required. We presumed that this condition emerged because of the delayed correction of coagulation by transfusing fresh-frozen plasma and vitamin K.Normally, conservative management is considered the first-line therapy, which constitutes immediate cessation of any anticoagulant, that is, correcting coagulation parameters by infusing fresh-frozen plasma and vitamin K, followed by bowel rest and total parenteral nutrition. However, the timing of starting this treatment (between 48 h and 2 months) was not described clearly.In conclusion, AT can lead to ISH, and the associated clinical symptoms can be diagnosed with barium enema, ultrasonography, abdominal CT, or endoscopy. Further, EUS or EUS-FNA can be performed to differentiate diagnosis of some similar or atypical lesions. Conservative management and surgical intervention are traditional treatment strategies. To avoid unnecessary surgery, EUS and EUS-FNA procedures should be recommended for unresolved hematoma or untreated intestinal obstruction in patients with stable vital signs.Conceptualization: Wei-Hua Yu.Data curation: Wei-Hua Yu, Chao Feng, Tie-Mei Han, Shun-Xian Ji, Lan Zhang.Formal analysis: Yi-Yang Dai.Investigation: Wei-Hua Yu.Methodology: Wei-Hua Yu.Supervision: Wei-Hua Yu, Yi-Yang Dai.Validation: Wei-Hua Yu.Writing – original draft: Wei-Hua Yu.Writing – review & editing: Wei-Hua Yu.


Dens evaginatus is a dental developmental anomaly that arises due to the folding of the inner dental epithelium that leads to the formation of an additional cusp or tubercle on the occlusal surface of the affected tooth. This accessory tissue projection may carry with it a narrow and constricted pulp horn extension. Occasionally, the tubercle easily fractures, thus leading to microexposure of the pulp horn and eventual pulp necrosis. Often, the pulp necrosis occurs at a time the root development of the affected tooth is incomplete. Apexification with calcium hydroxide and mineral trioxide aggregates has been the mainstay of treatment options before root canal obturation in immature nonvital permanent teeth. Lately, regenerative endodontics (maturogenesis) is becoming one of the preferred treatment modalities to manage such teeth. The current case highlights the possibility of a bioceramic material  which supposed to provide apical root closure (apexification) and could also induce continuation of root growth (maturogenesis). Dens evaginatus (DE) is a rare dental anomaly that is common in 1–4% people of Asian descent and mainly observed in the premolars. The mandibular premolars are five times most likely to have this developmental anomaly than the maxillary premolars . OehlersOne of the major clinical issues related to DE is the possible traumatic occlusion of the adjacent erupted tooth in contact. The traumatic forces may cause either attrition or fracture of the tubercle, thus resulting in inevitable pulp exposure . Pulp exThe current case describes the management of a nonvital immature mandibular right second premolar tooth with DE that developed pulp necrosis and symptomatic apical periodontitis. Although apexification was performed successfully with a newly available bioceramic root repair material , the use of this material also resulted in the unexpected continued root growth (maturogenesis).A healthy 11-year-old girl was presented to the National University of Malaysia (UKM) Paediatric Dental Clinic with a referral for further management of pulp necrosis of an immature lower right second premolar (tooth 45), secondary to the fractured tubercle of dens evaginatus. Two weeks earlier, she had treatment at a general dental clinic for pain related to tooth 45. Tooth 45 had spontaneous and lingering pain following cold and thermal stimuli. The tooth was diagnosed to have symptomatic irreversible pulpitis, and root canal therapy was initiated. The canal was accessed, and pulp extirpation performed before the placement of intracanal nonsetting calcium hydroxide by the general dental practitioner (GDP).At the time of current assessment, her tooth-related symptoms had completely resolved. General oral examination showed the presence of generalised mild gingivitis with a basic periodontal examination (BPE) score of 1 in all sextants. The patient's oral hygiene was fair with a plaque score of 30%. The patient is still in her mixed dentition with the presence of the primary maxillary canines. Her upper dental arch was well aligned, and mild crowding of anterior teeth was noted in the lower arch. Tooth 45 has an occlusal glass ionomer dressing of the access cavity made for the pulp extirpation earlier by the GDP . Cold anOn the day of initial assessment, tooth 45 was isolated with rubber dam after infiltration of local anaesthetic solution . Pulp chamber was reentered through the previously prepared access cavity. The root canal was exposed and irrigated with saline. After that, the canal was dried with paper points and the tooth working length was estimated with a K-file No. 60. A working length, 2 mm short of the apical opening, was determined (17 mm). The canal was gently prepared with the K-file No. 60 and then irrigated with a copious volume of 1.5% sodium hypochlorite (NaOCI). After drying the wet canal with paper points, nonsetting calcium hydroxide was placed into the canal and the access cavity was double sealed with Cavit™ 3M, USA, and glass ionomer cement (GIC) .Two weeks later, the tooth was reassessed for any signs and symptoms of infection. The tooth was no longer tender to percussion, and there was no indication of infection-related signs and symptoms. After isolation with a rubber dam, the root canal of tooth 45 was reaccessed and irrigated with a copious volume 1.5% NaOCl to remove the nonsetting calcium hydroxide. Then, the canal was irrigated with sterile water and dried with paper points. Subsequently, the canal was irrigated with 17% EDTA  for a minute and dried with paper points. Finally, under the guidance of a dental operating microscope , the apical region was filled using the EndoSequence  material up to 4 mm thickness to create an apical seal . The oriOnce again, the root canal was reaccessed two weeks later, irrigated with 1.5% NaOCl, and dried with paper points. Next, the dried canal was obturated with thermoplasticised gutta-perca using the Obtura III Max System (Obtura Spartan® Endodontics) . After tFollowing the obturation, tooth 45 was reviewed at three-month and six-month intervals. During both reviews, tooth 45 was asymptomatic. However, at the six-month review, a periapical radiograph of tooth 45 showed an unexpected finding. The apical root of tooth 45 continued to grow beyond the apexification level with a normal periodontal ligament space and lamina dura. No evidence of periapical radiolucency was noted . HoweverNumerous treatment modalities are available to treat teeth with dens evaginatus (DE) , 4, 5. HNecrotic teeth with immature roots provide significant challenges to clinicians especially with their wide open apices which do not allow successful canal obturation and thin dentine walls which are prone to fracture and compromised crown-root ratio . Over thThe regenerative endodontic technique (RET) is another treatment option that has received wide coverage in the literature regarding the management of nonvital permanent teeth in recent times. Although this technique is initially known as the vascularisation , other nIn the described case, we performed an apexification procedure on necrotic immature tooth 45 after discussing the various treatment options with the child's parents. In this procedure, the EndoSequence Root Repair Material (ERRM) was used to create an apical barrier. This material is a premixed bioceramic material which is composed of calcium silicates, zirconium oxide, tantalum oxide, calcium phosphate monobasic, and filler agents . The matThough the intended treatment for this patient is to create an apical barrier for root canal obturation, an unexpected finding was observed during the follow-up stages after the root canal obturation. We found that the root continued to grow and achieved its matured development. A normal lamina dura with an even periodontal space was noted around the apex of the tooth. One of the reasons for this occurrence is probably due to the osteoinductive property of the ERRM. The osteoinductivity of ERRM could have attracted the pluripotent cells from the surroundings to form preosteoblasts. The presence of preosteoblasts leads to the secretion of the bone matrix that later calcified into bone . Based oThe current case showed that ERRM is not only successful in creating an apical barrier in a nonvital immature permanent tooth for root canal obturation but also able to promote continued root growth. However, the potential of ERRM in fostering continued root growth on nonvital immature permanent teeth needs further clinical and research evidence.