Ingestion of fish bones leading to gastric perforation and inducing abscess formation in the caudate lobe of the liver is very rare.A 67-year-old man presented to our hospital with a 2-day history of subxiphoid pain. There were no specific symptoms other than pain. Laboratory tests showed only an increase in the number and percentage of neutrophils. Contrast-enhanced Computerized tomography (CT) of the abdomen showed two linear dense opacities in the gastric cardia, one of which penetrated the stomach and was adjacent to the caudate lobe of the liver, with inflammatory changes in the caudate lobe. We finally diagnosed his condition as a caudate lobe abscess secondary to intestinal perforation caused by a fishbone based on the history and imaging findings. The patient underwent 3D laparoscopic partial caudate lobectomy, incision and drainage of the liver abscess, and fishbone removal. The procedure was successful and we removed the fishbone from the liver. The patient was discharged on the 9th postoperative day without other complications.Liver abscess caused by foreign bodies requires multidisciplinary treatment. Especially when located in the caudate lobe, we must detect and remove the cause of the abscess as early as possible. Foreign bodies that perforate the gastrointestinal tract can penetrate to the liver and cause abscess formation, as in this case. When exploring the etiology of liver abscesses, we should investigate the general condition, including the whole gastrointestinal tract.The online version contains supplementary material available at 10.1186/s12893-021-01457-z. The presence of foreign materials on or in the liver is uncommon , 2. In gLiver abscesses have many causes, but fishbone perforation is a rare cause. In addition, fish bone insertion into the caudate lobe of the liver is even rarer. Because the caudate lobe is close to vital vascular structures, surgical resection is difficult and risky. It is considered the last area of laparoscopic liver surgery and was considered a difficult area for surgery in the past. Therefore, surgical treatment of liver abscess caused by a fishbone penetrating the caudate lobe of the liver is also a great challenge –10. In rSo, here we report a case of an abscess in the caudate lobe of the liver secondary to gastric perforation caused by a fishbone.A 67-year-old man was referred to the general practice department of our hospital due to subxiphoid pain. The patient was emergently admitted to our hospital. He had a 7-day history of eating fish. Before emergency admission, no special treatment was performed, except for an abdominal CT at the local hospital. He had a medical history of penicillin anaphylaxis. On emergency admission, his body temperature was normal. On the evening of admission, relevant laboratory investigations, such as routine blood tests, liver and kidney function, electrolytes, and inflammatory proteins were performed. Upon physical examination, the patient’s right upper quadrant was mildly tender to palpation without rebound tenderness. The white blood cell count was 18,150/μL (91.1% neutrophils). The C-reactive protein level was 125 mg/L. Serum bilirubin was normal. Glutamic oxaloacetic transaminase, and glutamic-pyruvic transaminase levels were 42 U/L, and 65 U/L respectively. On the first day after admission, considering that the patient had elevated inflammatory proteins and leukocytes, we performed a chest CT to see if there was a lung infection. Chest CT was also done as a routine test because of the Covid-19 pandemic. Chest CT revealed an infectious lesion in the upper lobe of the left lung with segmental atelectasis. On the second day after admission, we decided to perform a contrast-enhanced abdominal CT to better understand the patient’s abdominal cavity situation. The contrast-enhanced CT revealed two linear dense shadows in the gastric cardia, one of which penetrated through the gastric wall and was adjacent to the caudate lobe of the liver, accompanied by inflammatory changes in the caudate lobe. A patchy low-density shadow with enhanced edges measuring 41 mm × 38 mm was observed  examination to further confirm the presence of foreign material on the gastrointestinal tract, but the results did not reveal obvious leakage of contrast medium in the esophagus and stomach.Based on the patient’s history, signs and symptoms, and auxiliary examination results, we made a definite diagnosis of liver abscess caused by a fishbone piercing through the gastric wall and penetrating the caudate lobe.On day 4 of hospitalization, we performed a model reconstruction of the surrounding liver before surgery to better evaluate the general situation (Fig. On the 5th day after hospitalization, we decided to perform a 3D laparoscopic partial resection of the caudate lobe of the liver, incision and drainage of the liver abscess, and repair of the gastric perforation.Laparoscopic exploration revealed no significant ascites in the abdominal cavity, the liver was soft in consistency, and no significant sclerosis or tumor lesions were found. After exposing the caudate lobe, a sigmoidal fishbone could be seen outside the caudate lobe, and it was connected to the lesser curvature of the stomach Fig. .Fig. 5ThThe liver surface was covered with white pus. Inflammation, edema, and adhesions were observed in the lesser curvature of the stomach penetrated by fish bones, and perforation was observed. No other abnormalities were observed. Liver abscess, foreign body, and gastric perforation were diagnosed.During the procedure, firstly, we cut the ligamentum teres, the falciform ligament, the left part of the coronary ligament, and the left triangular ligament. Then we turned the left lateral lobe of the liver to the right, incised the lesser omentum close to the surface of the liver, exposed the caudate lobe, and pulled out the fishbone under direct laparoscopic vision. We also removed parenchyma with a radius of 1.5 cm around the fishbone until the abscess cavity was exposed. The pus in the abscess cavity was aspirated, and part of it was sent for bacteriological examination; at the same time, the lesser omentum around the caudate lobe, close to the lesser omentum at the lesser curvature and lower esophageal-cardia was resected posteriorly up to the anterior abdominal aorta, and repeated exploration along the lower esophageal-cardia revealed no leakage. After a complete examination of the condition in the abdominal cavity, the specimen was intactly removed. The first porta hepatis and inferior hepatic vein were not blocked during the operation. Postoperative histopathological examination revealed a large number of neutrophils infiltrated into some areas of liver tissue, along with necrosis, which was consistent with the changes observed in liver abscesses. The patient underwent iodine hydrography on the 4th day after surgery, and the examination results showed no significant contrast agent leakage in the esophagus and stomach. The patient had an uneventful postoperative recovery and was discharged on the 9th postoperative day (Additional file Inadvertent ingestion of fish bones is very common in China, especially in the elderly, children, and people with mental disorders. At the same time, people with diminished oral sensation are also prone to such conditions. The sharp edge of fish bones easily scratches the gastrointestinal mucosa and even causes peptic ulcers and perforation in severe cases. Although most of the ingested foreign bodies are excreted through the gastrointestinal tract within a week without other complications, unfortunately, gastrointestinal perforation occurs in less than 1% of patients . In thisAlthough our patient had no fever or nausea and vomiting, we diagnosed the patient’s condition as a liver abscess caused by the perforation of a fishbone into the caudate lobe based on the patient's self-report and enhanced CT findings.Endoscopy may be helpful if performed before foreign body migration and mucosal healing. Regarding the success rate of endoscopy, Jason Saltiel et al.  performeSo, we believe that oral contrast agents should be used as little as possible in the later diagnosis of non-traumatic abdominal pain. The perforation might have been difficult to detect using gastroscope under sedation and upper gastrointestinal radiography with iodinated water in our case. However, combined with the patient's chief complaint, enhanced CT, endoscopic ultrasonography, we were able to arrive at the diagnosis.It is worth noting that we do not use MRI to diagnose patients during the treatment process, but we believe that MRI can also improve the ability to diagnose accurately. In a case report of a hepatic abscess secondary to a rosemary twig migrating from the stomach by Karamarkovic et al. , MRI wasDuring laparoscopic exploration, a fishbone perforating the caudate lobe of the liver was observed under direct vision, and the omentum was wrapped near the caudate lobe. The lesser omentum was incised close to the surface of the liver, the caudate lobe was exposed with dissecting forceps, the fishbone penetrating the liver was pulled out under direct vision, liver parenchyma with a radius of 1.5 cm was removed until the abscess cavity was exposed. The pus was aspirated with an aspirator, some of it was sent for bacteriological examination, and a drainage tube was placed beside the abscess cavity. The abscess was flushed and drained, and the trocar hole was closed.During the operation, we should pay attention to the partial resection of the caudate lobe, the particularity of its location, and the complexity of its anatomical structure. The caudate lobe is surrounded by the three portae of the liver, it is adjacent to the inferior vena cava, portal vein, hepatic vein, and bile duct. The operating space is narrow, exposure is very difficult, intraoperative bleeding is difficult to control, and the operation risk is high. Advanced diagnostic techniques, familiarity with the anatomical characteristics of the caudate lobe, selection of the best surgical method and surgical approach, blood flow occlusion techniques, and prevention of postoperative complications are the basic requirements to guarantee a successful operation and prolonged survival –26.We use hepatectomy as the main treatment for patients, but there are other methods to treat foreign body-induced liver abscesses. For example, Horii et al.  treated Klebsiella pneumoniae is the most common pathogen that causes liver abscesses. However, in our case, no bacteria were found at the time of blood culture, but a culture of the purulent fluid from the liver abscess removed intraoperatively revealed growth of Streptococcus intermedius. This is the same genus that constitutes part of the normal bacterial flora of the human mouth, nasopharynx, and GI tract. We, therefore, suspect that oral, pharyngeal and GI streptococci have the potential to cause liver abscesses. So, the detection of these bacteria can also provide evidence that the liver abscess has developed from a foreign body that has penetrated the gastrointestinal wall to the liver.In this case, no foreign body was found by the gastroscope under sedation, which may be related to the small size of the foreign body. Compared with B-ultrasound and X-ray, CT or enhanced CT is a more sensitive method for diagnosing foreign bodies in the digestive tract, especially in coronal or sagittal reconstruction, with high density and spatial resolution. CT can not only find all foreign bodies, but it can also mark the position, shape, and adjacent relationship with the surrounding organs. Combined with the results of this endoscopic examination, it can be used to judge the operation track of foreign bodies in vivo, and it is an important reference for surgeons when formulating the surgical plan. However, there is a certain rate of missed diagnosis in imaging examination, and a negative result can not rule out the diagnosis of a foreign body, so we need to make a comprehensive judgment combined with the clinical findings.In conclusion, when a patient with a relatively special liver abscess is found, we need to listen carefully to the patient's chief complaints and ask carefully ask about the history. When upper GI endoscopy suggests the possibility of a perforation in the gastrointestinal tract, we need to be vigilant about infection of the abdominal organs, and enhanced CT scans should be performed to provide a sufficient basis for the treatment plan.Additional file 1: Chronology for diagnostic process.


Torpedo maculopathy (TM) is a rare congenital defect of the retinal pigment epithelium (RPE). The RPE is often evaluated clinically using fundus autofluorescence (AF), a technique that visualizes RPE structure at the tissue level from the intrinsic AF of RPE fluorophores. TM lesions typically emit little or no AF, but this macroscopic assessment is unable to resolve the RPE cells, leaving the organization of the RPE cell mosaic in TM unknown. We used fluorescence adaptive optics scanning laser ophthalmoscopy (AOSLO) to show here for the first time the microscopic cellular-level structural alterations to the RPE cell mosaic in TM that underlie the tissue-level changes seen in conventional clinical imaging. We evaluated two patients with TM using conventional clinical imaging techniques and adaptive optics (AO) infrared autofluorescence (IRAF) in AOSLO. Confocal AOSLO revealed relatively normal cones outside the TM lesion but altered cone appearance within it and along its margins in both patients. We quantified cone topography and RPE cell morphometry from the fovea to the margin of the lesion in case 1 and found cone density to be within the normal range across the locations imaged. However, RPE morphometric analysis revealed disrupted RPE cells outside the margin of the lesion; the mean RPE cell area was greater than two standard deviations above the normative range up to approximately 1.5 mm from the lesion margin. Similar morphometric changes were seen to individual RPE cells in case 2. Multi-modal imaging with AOSLO reveals that RPE cells are abnormal in TM well beyond the margins of the characteristic TM lesion boundary defined with conventional clinical imaging. Since the TM fovea appears to be fully formed, with normal cone packing, it is possible that the congenital RPE defect in TM occurs relatively late in retinal development. This work demonstrates how cellular level imaging of the RPE can provide new insight into RPE pathologies, particularly for rare conditions such as TM. Torpedo maculopathy (TM) is characterized by the presence of a well-circumscribed lesion of the macula featuring elements of hyperpigmentation and atrophy at the level of the retinal pigment epithelium (RPE) and named for its typical shape and orientation with respect to the fovea , 2. TM aIn TM, OCT sometimes reveals neurosensory retinal layers detached from the RPE, forming a cavity , 8. BaseIn recent years, OCT angiography (OCTA) has been used to evaluate flow profiles in the choriocapillaris and/or choroid in the lesions. Most of these studies have reported decreased flow in the choriocapillaris with vascular alterations at the site of the lesion , 14–16. in vivo in TM patients using any modality.Adaptive optics ophthalmoscopy (AOO) permits cellular level imaging of the retina , 20. RecGiven the well-circumscribed nature of TM, our hypothesis was that the transition from abnormal to normal cellular mosaics would be relatively abrupt, with abnormalities confined to the immediate vicinity of the lesion. Further, due to the non-progressive clinical course, we hypothesized that the demarcation between abnormal and normal photoreceptor and RPE mosaics would be approximately aligned, without one appearing to lead or lag the other in terms of distance from the lesion border.Clinical imaging included color fundus photography (Topcon/Canon), SLO/OCT , and a flood-illumination adaptive optics camera . Detailed OCT imaging parameters are presented in Adaptive optics scanning laser ophthalmoscopy imaging was carried out using a system that has been described in detail . BrieflyThe confocal images were used to co-register the weak signals in the AO-IRAF images using custom strip-based image registration software , permittPatients were recruited through the clinics of the UPMC Eye Center. Written informed consent was obtained from both subjects following an explanation of experimental procedures and risks both verbally and in writing. All experiments were approved by the University of Pittsburgh Institutional Review Board and adhered to the tenets of the Declaration of Helsinki. To ensure safe imaging, all light levels were kept below the ANSI laser safety limits  and werePatient 1, a woman in her early 30s was found to have a pigmented lesion in her right eye during pre-screening for laser refractive surgery that was diagnosed later as TM. Best corrected visual acuity (BCVA) was 20/15 in each eye. The patient had no history of retinal laser nor injections prior to our imaging. Patient 2 was a woman in her early 20s diagnosed with TM. Her BCVA was 20/20 in each eye. The left eye had received a single intravitreal bevacizumab injection 2 years earlier for neovascularization within the lesion. Her angiography images have been published previously .The first patient exhibited a typical torpedo-shaped lesion  in the tSince the second patient  was treain vivo morphology of both the cone and RPE cell mosaics in TM for the first time. These findings suggest that outside the TM lesion, RPE morphometry is relatively normal. However, near the margin of the lesion, we see marked changes in RPE morphometry characteristic of a disrupted cell mosaic. Within the TM lesion, we observed differences between the two cases that may reflect differences between type I and type II lesions. In patient 1  and also drafted the manuscript. ER, KD, and KV analyzed and interpreted the results. VS conducted the clinical imaging. JM and AE critically revised the manuscript for important intellectual content. All co-authors carefully reviewed the final manuscript.This research was supported by departmental startup funds from the University of Pittsburgh to ER. This work was also supported by the NIH CORE Grant P30 EY08098 to the University of Pittsburgh, Department of Ophthalmology, the Eye and Ear Foundation of Pittsburgh, NVIDIA GPU Grant Program and from an unrestricted grant from Research to Prevent Blindness, New York, NY, USA.Some aspects of this work include technologies that ER is an inventor on for patents that are owned by the University of Rochester . The remaining authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.All claims expressed in this article are solely those of the authors and do not necessarily represent those of their affiliated organizations, or those of the publisher, the editors and the reviewers. Any product that may be evaluated in this article, or claim that may be made by its manufacturer, is not guaranteed or endorsed by the publisher.


Bowel perforation as the first presentation of inflammatory bowel disease is rare and unusual in young patients. A previously asymptomatic 21-year-old Asian male presented with perforated small bowel secondary to previously undiagnosed Crohn’s disease. He underwent an exploratory laparotomy and subsequent small bowel resection and was commenced on mesalazine post-operation. He recovered well with subsequent regular follow-up with gastroenterology. The main management of Crohn’s disease is multidisciplinary in nature, and collaboration between different disciplines is inherent with the aim of reducing symptoms and maximizing patient quality of life. The perforation of bowel as the first presentation of inflammatory bowel disease is a rare occurrence reported in about 0.15–3% of the literature  and is eA 21-year-old Singaporean Chinese male was admitted for sudden onset generalized abdominal pain and one episode of non-bilious, non-bloody vomiting. He reported no history of fish bone ingestion, no diarrhoea and no loss of weight. He denied any history of joint pains, eye or dermatological symptoms. There was no family history of gastrointestinal disorders.9  on presentation.On presentation, his heart rate was 103, and he had a temperature of 37.7, with other vital signs within normal limits. On examination, he possessed a guarded abdomen, with generalized tenderness on palpation. His white cell count was 18.6 × 10A chest x-ray noted a small amount of subdiaphragmatic air. A computed tomography showed small bowel perforation at the distal ileum with scattered segmental wall thickening and mild surrounding fat stranding involving the transverse colon, terminal ileum and distal ileum as shown in The patient was commenced on intravenous ceftriaxone and metronidazole and underwent an exploratory laparotomy, small bowel resection and primary anastomosis.Intraoperatively, fat creeping of the small bowel was noted as seen in Histology of the specimen showed perforation into the mesentery and into peritoneal space, with granulomatous inflammation suggestive of Crohn’s disease. The acid-fast bacilli test for tuberculosis returned negative. The patient recovered uneventfully, and in view of the diagnosis of Crohn’s disease, was commenced on mesalazine 1 g twice daily on discharge.In this case, it is quite notable that the patient reported no symptoms until his first presentation to the hospital with perforated bowel. Free perforation of the bowel as first presentation of Crohn’s disease is rare with an incidence of 3.4%, with 86.1% of perforations involving the ileum . It is aThere are other diagnostic considerations for Asian countries. Intestinal tuberculosis can mimic the clinical manifestations of inflammatory bowel disease which can result in a misdiagnosis rate of 50–70%, and such a misdiagnosis can lead to complications once immunosuppressant medications are commenced to treat inflammatory bowel disease, as this may lead to reactivation or disseminated tuberculosis . Hence, When it comes to surgical management, between 70 and 90% of patients will require surgery during their lifetime, with up to 39% requiring repeated surgery. Indications for surgery are complications derived from strictures, intra-abdominal and perianal abscess and fistulae, bowel perforation, bleeding and malignancy. The main aspect of surgical management is to limit the amount of bowel resected as much as possible to avoid complications like short bowel syndrome . Young pAll authors are in agreement of the content of this manuscript.Written informed consent has been obtained from the patient for publication of details of their medical case and accompanying images. Ethics approval was not required.All authors discussed the results and contributed to the final manuscript. Dr Tze Tong Tey, Dr Fung Joon Foo and Dr Frederick Koh contributed to the management of the patient. Dr Jia Hui Lee wrote the manuscript. All authors approve the final version of the manuscript.The authors have no conflict of interest/competing interests to declare. This manuscript has not been published previously and is not under consideration elsewhere.This research received no specific grant from any funding agency in the public, commercial or not-for-profit sectors.


The popularity of robot‐assisted radical cystectomy over open radical cystectomy has been increasing because the former, a minimally invasive surgery, contributes to earlier recovery and shorter hospitalization. However, atypical recurrences may be more frequent after robot‐assisted radical cystectomy than after open radical cystectomy. We report a case of an atypical early recurrence of bladder cancer including the descending colon.A 70‐year‐old Japanese man underwent robot‐assisted radical cystectomy for muscle‐invasive bladder cancer. Four months later, he was hospitalized for severe anemia . Colonoscopy revealed a 4‐cm submucosal oozing tumor in the descending colon. Computed tomography revealed multiple recurrent lesions including recurrence in the descending colon, all of which disappeared completely after chemotherapy with six cycles of dose‐dense methotrexate, vinblastine, doxorubicin, and cisplatin.We encountered a rare case of an atypical recurrence of bladder cancer in the colon after robot‐assisted radical cystectomy. CRComplete responseCTcomputed tomographydd‐MVACdose‐dense methotrexate, vinblastine, doxorubicin and cisplatinORCopen radical cystectomyRARCrobot‐assisted radical cystectomyWe report a case of an atypical recurrence of bladder cancer in the descending colon after robot‐assisted radical cystectomy. Although there are several case reports of atypical recurrence after robot‐assisted radical cystectomy, recurrence in the colon has been rarely reported.Since first being reported in 2003, RARC has been presented as a potential alternative surgical procedure to ORC with the advantages of reduced blood loss, earlier postoperative recovery, and a shorter hospitaization.Here, we report a case of an atypical early recurrence of bladder cancer in the descending colon after RARC.A 70‐year‐old Japanese man was admitted to our hospital with a chief complaint of dysuria. Abdominal ultrasonography revealed a large bladder tumor, and cystoscopy revealed a nodular tumor filling the left half of the urinary bladder. CT revealed a cT2bN0M0 bladder cancer Fig. .He underwent transurethral resection of the bladder tumor and was diagnosed with high‐grade pT2 urothelial carcinoma with glandular differentiation. Left hydronephrosis developed 2 weeks later due to obstruction of the lower part of the left ureter. We immediately planned radical cystectomy following left percutaneous nephrostomy without neoadjuvant chemotherapy. He underwent RARC with urethral resection with an intracorporeal ileal conduit urinary diversion. The extra fourth arm was set on the left side of the patient, and 12 and 5 mm ports for bed‐side assistant were set on the right side of the patient. The lymphadenectomy was performed as a level of extended template including the common iliac, external iliac, internal iliac, and presacral lymph nodes. In an ileal conduit procedure, an approximately 25 cm segment of ileum was isolated and the distal ends of both ureters were anastomosed to the ileum by the Bricker technique. The total operative time and console time were 571 and 476 min, respectively. The estimate blood loss was 570 mL. Insufflation pressure was 10 mmHg. Histopathological examination revealed high‐grade pT2b urothelial carcinoma with glandular differentiation, INFβ, lymphatic invasion (+), vascular invasion (−), resected margin (−), and pN0 (0/55). The postoperative course was uneventful, and he was discharged approximately 1 month after RARC.Four months later, he was admitted to our hospital emergently for shortness of breath; his hemoglobin level fell to 5.1 g/dL. Gastrointestinal bleeding was suspected, and he underwent colonoscopy, which showed a 4‐cm submucosal oozing tumor in the descending colon Fig. . Tumor bEnhanced CT revealed intraperitoneal recurrence in the descending colon Fig. , massive2), vinblastine (3 mg/m2), doxorubicin (30 mg/m2) and cisplatin (70 mg/m2) every 2 weeks. He tolerated six cycles of the treatments without severe adverse events. CR according to the RECIST v1.1 was achieved  and 35 of 152 (23%) distant metastases and 6 of 150 (4%) and 4 of 152 (3%) local recurrences in the RARC and ORC groups, respectively.et al. compared 203 RARC cases and 278 ORC cases with a median follow up of 66 months at Mayo Clinic and reported that there were 29 of 203 (14.3%) and 53 of 278 (19.1%) distant metastases and 12 of 203 (5.9%) and 19 of 278 (6.8%) local recurrences in the RARC and ORC groups, respectively.Recurrences are of concern after radical cystectomy. Parekh 2 gas insufflation, surgical technique, and tumor biology are considered to relate to the occurrence of atypical recurrences.2 gas insufflation is essential in robot‐assisted urological surgeries. Experimental evidence suggests that pneumoperitoneum may increase the risk of intraperitoneal dissemination of cancer cells.2 gas insufflation could be the reasons of atypical recurrence in our case.Although recent studies have shown similar rates of atypical recurrence after ORC and RARC,et al. reported that dd‐MVAC treatment achieved a CR rate of 21% and a 5‐year survival rate of 21.8%.In this case, fortunately, we observed an excellent clinical and radiologic response to dd‐MVAC chemotherapy, with atypical recurrences such as intraperitoneal recurrence in the descending colon disappearing completely. dd‐MVAC chemotherapy is known to be more effective than classic MVAC, which has similar effectiveness to GC treatment,We report herein a case of atypical early recurrence of bladder cancer after RARC with multiple lesions including recurrence in the descending colon, local recurrence in the pelvic floor, and metastases to the extra‐pelvic lymph nodes and a port site, which disappeared completely after six cycles of dd‐MVAC treatment. This is the first report of an earlyrecurrence in the descending colon after RARC.Michio Noda: writing‐original draft. Masaki Nakamura: writing‐original draft, writing‐review & editing, supervision. Taketo Kawai: writing‐review & editing, supervision. Yusuke Sato: writing‐review & editing. Yuta Yamada: writing‐review & editing. Yoshiyuki Akiyama: writing‐review & editing. Daisuke Yamada: writing‐review & editing. Motofumi Suzuki: writing‐review & editing. Haruki Kume: writing‐review & editing, supervision.None of the contributing authors have any conflict of interest, including specific financial interests or relationships and affiliations relevant to the subject matter or materials discussed in the manuscript.Not applicable.All human subjects provided written informed consent with guarantees of confidentiality.Not applicable.


Very few pediatric cases of arachnoid cyst of ponto-cerebellar angle are described in the literature. Only 4 are described with hearing loss. It is a pathology which poses especially a problem of early diagnosis. In this paper we describe the management of a 16-year-old patient with an arachnoid cyst of the cerebellopontine angle with an isolated auditory deficit that was treated surgically. The follow up was marked by a Full recovery of hearing after surgical treatment. Arachnoid cyst of the cerebellopontine angle is rare in the pediatric population. early surgical management help to increase the chances of recovery. •Very few pediatric cases of arachnoid cyst of ponto-cerebellar angle are described in the literature. Only 4 are described with hearing loss.•This paper presents a 16-year-old patient with an arachnoid cyst of the cerebellopontine angle with an isolated auditory deficit that was treated surgically.•The case described in this article confirms the recommendation of Giordano et al. for rapid surgical management to increase the chances of recovery. Most often of slow and asymptomatic progression, these lesions may become symptomatic as a result of cyst enlargement or intracystic hemorrhage . These ctrans-tympanic drains and the last 10-year-old audiometry was normal.A 16-year-old boy, right handed, without Drug, family, and psychosocial history. Was brought by his parents to the ORL consultation after recently realizing isolated left hearing loss. During a game, he realized that he couldn't hear out of his left ear and he never realized it before. So it's hard to know if it was a sudden left-sided deafness or a slowly progressive deafness. His otological history includes the installation of several An audiometric balance by tonal and vocal audiometrics was performed, finding a left sensorineural deafness of medium intensity on low frequencies and deep apart from 1 KHz, with zero understanding. Analysis of auditory evoked potentials shows only a correctly recognizable peak 1 and the examination of automated otoemissions returns to a normal response. Finally, a vestibular assessment by VHIT and video-nystagmography finds respectively normal vestibulo-ocular reflexes for the 6 semi-circular channels and a slight left vestibular deficit estimated at 22% on caloric tests with a right nystagmic preponderance , Fig. 2.Neurological examination found conscious boy without a motor or sensory deficit, the balance was normal and no nystagmus. The rest of the neurological examination was normal.A brain CT SCAN was performed revealed a voluminous hypodense formation located on the left cerebellopontine angle, causing a mass effect on the bulboprotubercular territory. This process measures 34 mm of transverse development. It is 56 mm high .Fig. 3CTWe thought of an arachnoid cyst and epidermoid cyst so a brain MRI was performed reveals the presence, in years the left ponto cerebelled cistern, of an extra-axial signal mass similar to the CSF in the various weights. This mass not enhanced by contrast and does not have diffusion restrictions (high CDA such as CSF), which allows the diagnosis to be directed towards a large cyst arachnoid compressing the brainstem and the 4th ventricle without hydrocephalus . There iWe have retained the surgical indication The intervention was performed by our head chief department under general anesthesia. The patient was placed in a dorsal decubitus with a right tilt and rotation of the head to the right. A projection of a 2cm retro-sigmoid craniectomy respecting the sinuses. The craniectomy is performed until it reaches the limits of the sigmoid and transverse sinuses. A trident incision of the dura-mater is made. To reduce cerebellar traction, a first cyst int the left cerebellopontine cistern is opened to drain CSF. The beginning of the approach is carried out under microscope  until we saw the posterior wall of the arachnoid cyst. We then open it next to the acoustico-facial complex. The next steps of the surgery are performed under endoscopy 3D HD 4mm 30 . A sample of the wall is taken to perform an anatomopathological analysis. After suctioning the fluid, the walls of the cysts are carefully separated from the left vertebral artery, basilar trunk, antero inferior cerebellar artery, facial nerve and vestibulocochlear nerve in order to obtain optimal ablation. Endoscopy exploration of the cystic cavity identifies and opens several other cystic walls to the trigeminal and mixed nerves. Following this dissection, good communication with the cisterns is observed , Fig. 6.No bleeding occurred during this 90-min operation. Closure of the dura-mother and filling of the craniectomy with bone powder (autograft) and tissue glue (Tisseel). No post-operative CSF leaks are found.The patient spent one night in the intensive care unit before returning to the neurosurgery service. The cerebral CT-scan on the first postoperative day shows a clear reduction in the size of cystic formation of the left CPA. Impression confirmed by MRI of the fourth postoperative day which shows a good volume regression of the arachnoid cyst with a less marked mass effect on the brainstem and on the left acoustic facial nervous complex that is clearly visible on the posterior side of the cyst. Apparition of CSF flow artifacts in the cyst reflecting its communication with the cisterns of the posterior pit .Fig. 7A:The patient developed nausea and vomiting as well as mild photophobia from the second postoperative day which resolved following optimal intravenous hydration and a timely introduction of antiemetic drug . No abnormalities on neurological examination are shown postoperatively, no ataxia, dysmetry or adiadococynesis are noted during the clinical examination. Audiometric tests on the fourth postoperative day show a complete recovery of his hearing .Fig. 8PoThis case has been reported in line with the 2020 SCARE guidelines .2As their origin is still poorly understood, different hypotheses are considered. These hypotheses include a secretion of LCR through the cyst wall and a fluid accumulation secondary to a change in osmotic gradient. Arachnoid cysts generally do not show communication with subarachnoid spaces however a third hypothesis suggests the existence of a one-way valve system allowing communication between the cyst and sub-arachnoids spaces .The first reported cases of subarachnoid cysts of the cerebellopontine angle (CPA) made the subjects of a classification into 3 groups: Tumor Pseudo, Cystic and Adhesive ,11. WithOnly 18 pediatric cases of subarachnoid cyst of APC are described in the literature. Only 4 had hearing loss. In children, they have a prevalence of 2.6% with a preponderance of boy ,10. TheyThere are only a few cases described in the literature concerning arachnoid cysts operated in a pediatric population . The indHearing loss is a rare symptom, described only in 4 children of which only 2 had total recovery after surgery . Its onsThe use of 3D endoscopy  has limited the size of craniectomy, make us able to have a better visualization of neurovascular structures such as brain stem, left vertebral artery, basilar trunk, antero-inferior cerebellar artery, trigeminal nerve, facial, vestibulocochlear and mixed nerves and thus have a good visualization of Pacchioni oval foramen at foramen magnum . Its useOur case supports the idea that in children, a short period of time between diagnosis and neurosurgical management is a key factor in optimal clinical improvement.3Cysts of the ponto-cerebellar angle are rare in the pediatric population. The delay in surgical management after the onset and/or progression of symptoms is paramount. The case described in this article confirms the recommendation of Giordano et al. for rapid surgical management to increase the chances of recovery.The authors declare having no conflicts of interest for this article.None.Written informed consent for publication of their clinical details and/or clinical images was obtained from the patient. Ethical approval has been exempted by our institution.None.None.Marouane MAKHCHOUNE: Corresponding author and writing the paperUlysse CONEYS: writing the paperMichel TRIFFAUX: writing the paperMarie-Anne LABAISSE: Correcting the paperAnne DOYEN: Correcting the paperMAKHCHOUNE MAROUANE.The authors declared that this study has received no financial support.Not commissioned, externally peer-reviewed.


An epigastric hernia is a form of ventral hernia. Most of these contain preperitoneal fat or the omentum. Herniation of intra-abdominal organs, a rare form of rectus sheath midline herniations, is seldom seen, and incarcerations in these cases are rare. A 56-year-old male presented with complaints of irreducible epigastric swelling. Thorough clinical examination and imaging studies revealed that the patient had an epigastric hernia. An intraoperative image showed that the small bowel had herniated through the epigastric defect and was obstructed; however, the small bowel was viable. The contents were reduced after enlarging the constricting ring, and anatomical repair with mesh reinforcement was done. Postoperatively, the patient had an uneventful recovery and was discharged. The most common forms of external hernias are inguinal, umbilical, femoral, and incisional . Among tA 56-year-old male presented with complaints of a slow, progressive swelling in the upper abdomen for the last one month. He developed abdominal pain, vomiting, and obstipation for the last two days. The abdominal pain was colicky in type, in the epigastric region, moderate in intensity, with aggravation on food intake. The patient had five episodes of bilious vomiting per day, containing food particles, and was non-blood stained. On examination, the patient’s vitals were stable with tachycardia of 110 bpm and blood pressure of 110/70 mmHg. Abdominal examination showed a 1.5-cm epigastric defect, with the herniated small bowel forming a swelling of size 5 cm x 6 cm, irreducible, without any cough impulse (Figure 3. The abdominal X-ray suggested dilated small bowel loops with features of intestinal obstruction (Figure Blood tests showed normal hemoglobin, liver function, and renal function. Blood counts showed mild leukocytosis at 13,450 cells/mmn Figure .The patient was diagnosed to have incarcerated epigastric hernia with intestinal obstruction. Hence, the patient was immediately taken for exploratory laparotomy. An intraoperative image showed extensive adhesions between the prolapsed contents and the edge of the defect in the abdominal wall Figure .The herniated loops were released from each other, and the constricting ring was released by extending the incision. The bowel was released from the hernia site and was viable, without any evidence of gangrenous changes. The contents were reduced, and primary closure of the hernia defect was done and reinforced with a non-absorbable mesh. Postoperatively, the patient improved without any complications.The prolapse of tissue through a defect in the linea alba between the xiphoid process and the umbilicus is called an epigastric hernia. Anatomical involvement has seen predominance in the immediate 3 inches above the umbilicus while the incidence becomes rarer proximally. Epigastric hernias near the xiphoid process are rarely reported in the medical literature . It is mThese hernias are usually small in size, with preperitoneal fat as the usual content. The prolapse of intra-abdominal viscera is a rare occurrence, as the defect caused by the perforator vessels is usually very small. This is one of the oldest theories proposed by Moskowitz . It has Most of the patients remain asymptomatic for long periods, presenting with complaints of periodical pricking pain over the swelling, increasing food intake, and dyspepsia . This maAn incarcerated epigastric hernia is an emergency that most commonly requires immediate surgical exploration to relieve the obstruction. Although the progression from an incarcerated hernia to gangrene can be as fast as within six hours, early intervention can prevent gangrene of the contents. Healthy contents can be reduced into the abdomen, and a primary anatomical repair of the rectus sheath can be done. Depending on the preoperative state of the patient, secondary enforcement with mesh augmentation can be planned in the same setting or as a second surgery .Moreover, in the case of gangrenous contents, urgent, rapid exploratory laparotomy is usually followed by resection of the gangrenous segment. Complicated epigastric hernias increase the risk of local complications, including surgical site infection, recurrence, and mesh infection, which can increase the morbidity of the patients ,11. In oA complicated epigastric hernia is a rare phenomenon for which clinicians need to maintain a high risk of suspicion. Diagnosis is usually clinical, and imaging may be performed in doubtful cases. Due to the rapid deterioration, immediate diagnosis and prompt surgical intervention are the approaches of choice. This helps not only in avoiding unnecessary complications and the delay in recovery usually associated with resection and anastomosis of the gangrenous bowel segment but also in the expeditious recovery of the patient.


Clinicians should consider uterine torsion as a differential diagnosis for acute abdominal pain in women with scoliosis. Scoliosis may have led to an abnormal pelvic structure, making it easier for pelvic organs to twist. Clinicians should consider uterine torsion as a differential diagnosis for acute abdominal pain in women with scoliosis. Scoliosis may have led to an abnormal pelvic structure, making it easier for pelvic organs to twist. Physical examination revealed a palpable mass in the lower abdomen. X‐ray revealed severe scoliosis Figure . ContrasBased on the preoperative diagnosis of either side of ovarian torsion, total hysterectomy and bilateral salpingo‐oophorectomy were performed. Intraoperatively, the uterus appeared mostly necrotic and twisted by 180º Figure . The uteUterine torsion, defined as >45º twisting of the uterus on its long axis, is rare.The authors would like to thank the patient for giving consent.None declared.KK provided case information, references, and wrote the manuscript. FK and IY provided case information and clinical images.Standard ethical approval was obtained.Published with written consent of the patient.


Cavernous hemangiomas are benign malformations of vascular origin, usually well circumscribed and slow to grow. These lesions can be asymptomatic, being discovered unintentionally in imaging exams or symptomatic, indicated mainly by the presence of proptosis, diplopia, and visual disturbances by optic nerve compression. The complementary exams involve computed tomography associated with contrast, color Doppler, magnetic resonance, and angiography. Treatment can be conservative or surgical depending on the case, and the open therapy usually involves lateral, supraorbital, transconjunctival, transantral, pterional, transnasal, and extradural endoscopic orbitotomy. The present study aimed to report a recurrent case of hemangioma in the orbital cavity signaled by ocular proptosis, hyperemia, and ocular pain.The lesion was achieved through the Weber-Ferguson access with zygomatic osteotomy and preservation of the infraorbital nerve. The excision of the lesion was performed, and the previously displaced fragments were fixed with 1.5 mm mini plates. The patient has a chance of progressing with visual impairment due to considerable manipulation of the optic nerve and is being followed up.The reported case showed a successful diagnosis and therapeutic conduct, remaining now in the evolution and follow-up scenario. Vascular malformations can be classified into four categories: arteriovenous malformations; cerebral cavernous malformations, also known as hemangiomas or cavernomas; venous angiomas; and finally capillary telangiectasias. Among these, hemangiomas occupy the second place with the highest incidence, second only to arteriovenous malformations.It is a benign lesion with slow growth, composed of an endothelial proliferation that appear during angiogenesis with irregular sizes.It is estimated that 80% of orbital hemangiomas are housed in the intraconal compartment (between the Tenon’s capsule and the extraocular muscles) usually in the lateral direction.It is worth pointing out that they are usually asymptomatic, but patients with this tumor may present visual symptoms, such as ptosis and vision deficits by compression of the second cranial nerve pair—the optic nerve—in addition to psychological and aesthetic impairment.Thus, the current study aimed to report a case of a hemangioma diagnosed in the orbital cavity in a 40-year-old patient.A 40-year-old female patient presented to the head and neck surgery service with a report of protrusion of the right eyeball in early 2019. A magnetic resonance imaging (MRI) exam was requested to complement the diagnosis, showing a tumor mass positioned in a retro-orbital and inferior-medial form . The caHowever, in September 2019, the progressive exophthalmos was observed, being indicated as a potential sign of recurrence . Thus, This fact was accompanied by signs of evolution represented by ocular proptosis, hyperemia, and ocular pain of progressive character indicating the need for surgical intervention. Thus, we opted for the Weber-Ferguson approach, with tissue divulsion and exposure of the zygomatic bone . The viA complex resection was performed because it is a lesion occupying the entire posterior orbit and involvement of the optic nerve and eye movement muscles . After Cavernous orbital hemangiomas (HCOs) are benign circumscribed vascular malformations and with a slow growth development 1–5) and may in some cases present acutely.–5 and maDas et alAs for the etiology of HCO, it is understood that the most common cause may result from an underlying orbital vascular anomaly: acute orbital hemorrhage from normal vessels after trauma and valsalva maneuvers in compromised patients, or from the presence of venous-lymphatic malformations, and arteriovenous malformations, in addition to neoplastic vessels (tumors).Cavernous hemangiomas of the orbit can be asymptomatic, being discovered accidentally on imaging studies, or symptomatic, with the most common manifestations being proptosis, pain, diplopia, and visual disturbances due to compression of the optic nerve.To confirm possible diagnostic hypotheses, it is interesting that the surgeon uses complementary exams, with CT, associated with the use of contrast dye, the most common method.These exams can assist in the proper establishment of the pathology, thus ruling out other diseases of orbital cavernous malformations. Some types of cysts, vascular and peripheral nerve injuries, tumors of the optic and meningeal nerve, and capillary hemangioma or lymphangioma can be considered differential diagnoses, which must be carefully analyzed to establish an appropriate therapeutic approach.It is noteworthy that HCOs are generally identified because they are caves lined with blood-filled endothelium, which differentiate them from other orbital vascular tumors, such as capillary hemangiomas, hemangiopericytomas, hemangioendotheliomas, and angiofibromas.After the diagnosis is established, the surgeon must plan the patient’s treatment plan according to the peculiarities of the case. Treatment may be surgical or not, and this will depend on the location and complexity of the lesion. The most common surgical approaches are lateral, supraorbital, transconjunctival, transantral, pterional, transnasal, and extradural endoscopy.The HCOs located in the medial orbital compartment, lower, and at the orbital apex, are indicated for transnasal endoscopic approach. This approach generates orbital decompression, even in cases with partial removal of the lesion.Lenzi et alNonsurgical methods are recommended for small, asymptomatic, and slow-growing masses.The recurrence rate is rare after surgical excision, but it has been reported in the literature, 5 as in this case, in which the patient presented a recurrence of the lesion after the first excision.Surgical treatment of HCOs is indicated only for symptomatic patients with proptosis, diplopia, pain, and reduced visual quality.Based on its rate of achievement and success, lateral orbitotomy is considered the most common surgical approach, as it provides an excellent exposure of tumors located in the upper, lateral, or lower compartment of the orbit.In this perspective, it is observed in the literature the report of several cases of orbicular cavernous hemangioma between men and women, aged 20 to 60 years old, who presented similar signs and symptoms and underwent imaging tests and surgical treatment for injury removal and risk reduction.Among these authors, Yan and WangRegarding the injury recurrence in the case reported here, Choudhri et alOrbicular cavernous hemangioma is considered a common diagnosis for adult patients with evolution of proptosis and presence of an orbital mass. The surgical approach must be individualized based on the peculiarities of each case. The treatment will be successful due to the degree of fusion between the lesion capsule and important structures, such as motor and sensory nerves and muscles. If the lesion is not completely removed, the risk of recurrence is increased, and the patient must have its evolution monitored. In the present case report, the patient had a successful diagnosis and therapeutic conduct, remaining now in the evolution and follow-up scenario.


We present a case of dysgerminoma of the right adnexa with an infiltration to the right wall of the uterus and a metastasis of para‐aortic lymph node. A complete surgical staging may be usually needed in cases of ovarian dysgerminoma. Fertility‐sparing surgery will be allowed only for young women with limited tumor spread as results of detailed examination. A 49‐year‐old nulliparous woman visited our hospital due to abdominal fullness. Her general condition was good. Magnetic resonance imaging revealed a 10.5 × 8.5 × 6.7 cm solid, enhancing right ovarian mass Figure . A complIn the current case, we suspected dysgerminoma preoperatively. Therefore, lymphadenectomy may have been avoided because dysgerminomas usually have excellent prognosis after a simple salpingo‐oophorectomy especially in cases of unilateral tumor without capsular invasion or spread due to their excellent response to chemotherapy.All authors declare no conflict of interest relevant to this article.RK (primary author) analyzed the data and wrote the manuscript. SS involved formulating the concept of the study, analyzed the data, and drafted the manuscript.Prior to the case study, written informed consent was obtained from the patient. This case report was approved and licensed by the Ethics Committee of the Nippon Medical School.Written informed consent was obtained from the patient.


Solitary fibrous tumor (SFT) is a relatively rare mesenchymal tumor that mainly affects adults. Its prognosis is good after curative resection, but distant recurrences after 10 years or longer have been reported. Recurrent SFT usually arises as a local lesion; distant metastasis is rarely reported. Here, we report lung metastases that recurred a decade after excising a retroperitoneal primary SFT.A 44-year-old woman had an SFT resected from her right retroperitoneum at our hospital. Ten years later, at age 54, she underwent a lung resection after CT showed three suspected metastases in her left lung. All three were histologically diagnosed as lung metastases from the retroperitoneal SFT. However, whereas the primary SFT had 1–2 mitotic cells/10 high power fields (HPF), the metastatic lesion increased malignancy, at 50/10 HPF.Patients who have had resected SFTs should be carefully followed up, as malignancy may change in distant metastasis, as in this case. Solitary fibrous tumor (SFT) is a spindle-cell neoplasm with a varied presentation. It is a rare disease, occurring in only 2.8/100,000 people. SFTs usually develop in the pleura, but 30–40% of SFTs arise in extra-pleural regions [A 44-year-old woman presented with a chief complaint of abdominal distension. Her primary physician found a retroperitoneal tumor on palpation. She had no medical history and no comorbidities. Laboratory tests showed no significant abnormalities in total blood count, inflammation, liver function, renal function, electrolytes, or coagulation. Various hormone tests were all negative. Computed tomography (CT) showed a 16 × 16 × 10-cm tumor in the right retroperitoneum, which ventrally displaced the duodenum and right kidney (Fig. No distant metastases were found, and the retroperitoneal mass was curatively resected by laparotomy. The tumor was detachable from the right adrenal gland and other organs, and was judged to be a retroperitoneal tumor. The specimen was 20 cm in size, elastic with soft tissue (Fig. The tumor was diagnosed as benign, but because of its huge size, CT scans were performed every 6 months for 5 years. Ten years after resection, a CT scan incidentally revealed metastasis lesions. A CT scan found three nodular shadows in her left lung, including a 15-mm nodular shadow on the lower left lobe, and contrast effects indicating 9-mm and 8-mm margins on the upper lobe Fig. a–c. TheySFT is an uncommon soft-tissue neoplasm that was first reported as a type of pleural neoplasm in 1931 . It occuMicroscopically, SFTs show multiplying spindle cells with a patternless arrangement, and hemangiopericytoma-like appearance with prominent vascularity . Among iAlthough SFTs are typically benign, they can become malignant, especially if they grow to a large size or repeatedly recur , 8, 9. APatients who have had resected SFTs should be carefully followed up, as malignancy may change in distant metastasis, as in this case.


Hepatocellular carcinoma (HCC) with the concurrent occurrence of primary hepatic neuroendocrine carcinoma (NEC) of the liver is extremely rare. Preoperative diagnosis of HCC combined with NEC is very difficult. Here, we presented a case report of HCC combined with NEC. A 33-year-old male was admitted to our hospital due to focal liver lesion. To further the diagnosis, he received laboratory tests, conventional ultrasound, contrast-enhanced ultrasound (CEUS), and magnetic resonance imaging (MRI). Grayscale ultrasound showed a hypoechoic nodule with peripheral hypoechoic halo and central small patches of anechoic area in the hepatic segment VI, and the liver background was cirrhosis. In the CEUS, the solid component of the nodule was rapidly homogeneous hyper-enhancement in the arterial phase. Then, the enhancement of the nodule was washed out slowly and gradually; the nodule presented heterogeneously mild enhancement in the portal phase, and slight hypo-enhancement was showed in the late phase. The hypo-enhanced area was mainly located in the surrounding space. Meanwhile, there was a small piece of non-enhanced area within the lesion center in the whole three-phase imaging. MRI showed a lobular contoured mass in segment VI. The patient underwent middle liver resection, splenectomy, and cholecystectomy. The pathological diagnosis was a HCC with NEC. At the time of the preparation of this manuscript, the patient has been alive without recurrence or distant metastases for 6 months since the surgery. We mainly focus on the ultrasound imaging characteristics, especially its enhancement manifestations on CEUS. In this report, since this article is a case report, which is based on the clinical information of the patient and does not involve the patient's privacy, informed consent is not necessary. In addition, the patient agreed to publish the case. To the best of our knowledge, this report is the first to describe the CEUS patterns of the HCC combined with NEC. Herein, we report a case that provides novel insights that will improve clinicians' awareness of the clinical and ultrasound manifestations of this mixed tumor, resulting in improved diagnosis, treatment, and outcomes. Two or more new tumor tissue structures may be detected simultaneously in several organs, which is called a mixed tumor. In the liver, most of the mixed carcinomas consist of a combination of hepatocellular carcinoma (HCC) and cholangiocarcinoma –3. HowevA 33-year-old male, who claimed to have had a 5-year history of liver cirrhosis with antiviral treatment and liver protection drugs, was admitted to our hospital due to a “liver space-occupying lesion” for the past 2 weeks. Physical examination revealed abdominal flatness and no tenderness point, but the spleen was swollen. Laboratory studies detected infection by hepatitis B virus and almost normal liver function. However, serum examinations detected a level of alpha-fetoprotein (AFP) of 403 ng/ml. Then, the patient underwent an abdominal ultrasound examination by a Resona7 ultrasound system  equipped with an SC6-1U (1–6 MHz) and L9-3 (3–9 MHz) transducer. The conventional B-mode ultrasound image showed a malformation of the liver; the capsule of the liver was not smooth, and the echo of the liver parenchyma was rough and heterogeneous. Meanwhile, a hypoechoic nodule with peripheral hypoechoic halo, located in the hepatic segment VI, was found. The lesion had an approximate size of 2.7 cm × 2.2 cm, with an almost regular shape and slightly clear margins; small patches of anechoic area were detected within the tumor . Color DSoon afterward, middle liver resection, splenectomy, and cholecystectomy were performed. Severe liver cirrhosis changes were found during the operation. An intraoperative ultrasound revealed that the nodule was located in segment VI, extending to segment V, with an approximate size of 2.7 cm × 2.0 cm. No tumor changes were observed in the rest of the liver. Microscopic examination revealed that the tumor consisted of two components: a HCC and a NEC . ImmunohThe coexistence of HCC and NEC is rare as compared with the mixed type of HCC with cholangiocarcinoma in the liver, which is mainly due to the relatively low incidence of primary hepatic NEC. With the aim to investigate more comprehensively and understand better the clinical characteristics, imaging performance, and treatment, here, we present a case of a liver tumor with classical HCC combined with NEC. In this case, the patient was young, male, and had a history of hepatitis B, which is consistent with previous evidence –6. To suThe precise diagnosis of this rare mixed tumor is based predominantly on postoperative pathological examination findings. The patient in the case was diagnosed with HCC of the liver complicated with NEC, established by postoperative pathology. The pathological diagnosis was based on the following findings: (1) morphologically, the tumor tissue was composed of two components, with HCC morphology and with NEC morphology. This tumor was a HCC with the features of a small NEC. The two types of tumor cells were separated by wide fibrous band, with no transition areas observed under a microscope; hence, a single disease could not explain the various morphologies; (2) immunohistochemical results: strongly positive HCC markers, HEPA(+) and GPC-3(+), and positive neuroendocrine markers, CgA(+), Syn(+), and CD56. (3) The tumor size was 2.5 cm ×2 cm, with most of the area occupied by the hepatocellular carcinoma tumor, accounting for ~98% of the whole tumor area. (4) The results of abdominal MRI did not support the diagnosis of metastasis to the liver from other sites. In addition, regarding the genetic analysis results of this mixed tumor, both tumor components contained genetic mutations in the CTNNB1 gene (S33F located in exon 3), which were shared in the PD-1, PGP, and SMO of this gene mutation , similar cystic changes within the tumor are rarely found –12. TherThe prognosis and treatment of HCC accompanied by NEC are unclear due to the limited number of cases reported. Our analysis of the literature found that the presence of NEC components implicates the possibility of aggressive behavior of the mixed-type tumor , 16, 17.In summary, although a mixed HCC and NEC tumor is a rare condition, in patients with chronic liver disease, the rare possibility of HCC–NEC needs to be considered in the differential diagnosis, regardless of the presence of cirrhosis even if diagnostic clinical signs are not manifested and imaging features are lacking. Although the final diagnosis is based mainly on pathological and immunohistochemical examinations, ultrasonography, including CEUS, is widely used for detection of liver lesions.Our case is important as it is the first to highlight the value and utility of CEUS for diagnosis in patients with HCC tumor combined with NEC. Our report provides novel insights that can serve as a basis for further research and will improve clinicians' awareness of the clinical and ultrasound manifestations of this mixed tumor, resulting in improved diagnosis, treatment, and outcomes.The raw data supporting the conclusions of this article will be made available by the authors, without undue reservation.Written informed consent was obtained from the individual(s) for the publication of any potentially identifiable images or data included in this article.HW collected, conducted, and interpreted the patient data and contributed significantly to the writing of the initial manuscript. WL revised the paper and had primary responsibility for the final content. ZW performed and analyzed pathological data. YL contributed to the conception of the study. DY was the major contributor in coordinating patient care and helped perform the analysis with constructive discussions. The final manuscript was read and approved by all authors.The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.All claims expressed in this article are solely those of the authors and do not necessarily represent those of their affiliated organizations, or those of the publisher, the editors and the reviewers. Any product that may be evaluated in this article, or claim that may be made by its manufacturer, is not guaranteed or endorsed by the publisher.


Schwannomas are rarely seen in the foot and ankle and mostly arise from the plantar aspect. Dorsal foot schwannomas are not commonly reported. We describe the case of a 25-year-old man who had a painful swelling on the dorsum of his right foot, which resembled a ganglion cyst on clinical examination and ultrasonography findings. Persistent symptoms prompted the need for an excisional biopsy, which revealed a benign schwannoma arising from the medial dorsal cutaneous nerve. Complete and careful excision relieved the symptoms without causing any distal neural deficit. Thus, schwannomas can mimic as a ganglion cyst. Excisional biopsy of such swellings, if symptomatic, helps in confirming the diagnosis, and provides a good functional outcome. Peripheral nerve sheath tumors (PNSTs) are relatively rare tumors, arising from the sheath of cranial nerves, spinal nerves, or peripheral nerves. Among those, schwannomas, also known as neurilemmomas, are one of the most common benign neoplasms, arising from the Schwann cells . SchwannA 25-year-old male presented with complaints of a small painful swelling over the dorsum of the right foot for the past one year. There was no antecedent history of any trauma/ similar swellings elsewhere in the body neither was there any history of an increase in the size of the swelling. Aspiration was attempted from the swelling in another hospital but was not successful. Examination revealed a 2-cm, mobile, non-compressible, soft, tender swelling over the mid-dorsum of right mid-foot just proximal to the level of tarsometatarsal joints. Tinel’s sign was negative giving a clinical suspicion of a ganglion cyst. Visual analog score (VAS) was 6/10. There was no gait abnormality observed. Ultrasonography of the right foot demonstrated a soft tissue hypoechoic mass with posterior acoustic enhancement simulating a ganglion cyst; however, continuity of adjacent nerve fibers also gave a differential diagnosis of PNST. In view of the conflicting ultrasonographic and clinical findings, unavailability of MRI, and persistent symptoms, an excisional biopsy was planned. Upon giving incision in the superficial fascia, a smooth shiny bulbous mass was visualized in continuity with medial dorsal cutaneous nerve; careful excision of the mass was done maintaining the continuity of the nerve fibers. The clinical and intra-operative pictures of the swelling have been described in Figures The histopathology revealed an encapsulated mass showing an admix of myxoid hypocellular (Antoni B) and hypercellular areas (Antoni A) with nuclear palisading (Verocay bodies), confirming the diagnosis of benign schwannoma Figures -2C.The patient’s symptoms were completely resolved after the surgery, and he resumed his normal activities from the next day. There was no neural deficit or any other complication noted after the excision. At the last follow-up of one year, the patient was pain-free (VAS 0) with no evidence of recurrence. Consent was obtained from the patient for the publication of this manuscript.Schwannomas are a histological subtype of PNSTs, constituting 5% of all benign soft-tissue neoplasms . Foot anPeripheral nerve schwannomas usually present in the third-fourth decade with an equal predilection for male and female patients . In the Ozdemir et al. described 196 primary bone and soft-tissue tumors involving foot and ankle at a tertiary center over 10 years, in which only two cases of benign schwannomas were reported . In anotSchwannomas should be kept as a differential in all dorsal midfoot swellings. The clinical presentation of such swellings may mimic that of a ganglion cyst. Excisional biopsy of such swellings, if symptomatic, helps in confirming the diagnosis and provides a good functional outcome.


The supracondylar process is a beak-shaped bone spur arising from the anteromedial area of the distal humerus and in the majority of cases, it is connected to the medial epicondyle with a band of connective tissue which is known as ligament of Struthers. The complex of bone spur and ligament creates a ring that may compress the median nerve causing soreness and paresthesia of the hand and fingers. We present a rare case of bilateral supracondylar process compressing the neurovascular bundles in both arms and causing simultaneous bilateral upper limb pain, numbness, and weakness in an otherwise healthy young child. Surgical excision of the bone spurs and release of Struthers' ligaments provided immediate pain relief and complete resolution of symptoms. Three years after the index surgery, no limitations of activities or signs of recurrence were reported. Median nerve compression neuropathy in a child or a young adult should always raise the suspicion of the presence of supracondylar process particularly when bilateral symptoms exist. Although there are limited data about the incidence of recurrence and the extent of bone resection, the supracondylar process should be excised together with the overlying periosteum to minimize the theoretical risk of local regrowth. The supracondylar process is a well-documented anatomical variant with an incidence between 0.4% and 2.7% -3. It isA 12-year-old Caucasian male patient without previous trauma and negative medical history presented with a six-month history of progressive, subacute, burning pain in the volar aspect of his both distal arms and along the distribution of median nerve. Symptoms were initially apparent during sports and manual exertive activities but gradually were present at rest and even during the night. The patient also reported paresthesia and numbness of the hands and involved fingers during the night hours. On physical examination, there was marked tenderness at palpation on both anteromedial supracondylar arm regions and a Tinel sign was elicited on gentle percussion of the area bilaterally. No motor or sensory deficit was observed, and electromyography did not reveal any specific abnormality. Radiographs showed an anteromedial supracondylar bone spur in both humeri approximately 6 cm from the medial epicondyle. A radiolucent line was evident in the base of the left bone spur that indicated a stress fracture or incomplete fusion of the supracondylar process  derived from the exostoses and running distally was also recognized superficial to the neurovascular bundle which was found compressed and slightly deformed. Both osteocartilaginous exostoses were excised from their base with an osteotome and the attached Struthers' ligaments were also resected to release the median nerves and the adjacent brachial arteries Figure . Bone wax was not utilized to avoid the possibility of infection and foreign body reaction. Histopathological examination confirmed the benign character of the bone spurs, excluding malignancy. The postoperative course was uneventful, and the patient experienced immediate pain relief and complete resolution of symptoms. Three years after the index surgery, no limitations in his activities or signs of recurrence were reported. The disabilities of the arm, shoulder, and hand (DASH) score were zero in both upper limbs.Bilateral median nerve neuropathy due to compression by the supracondylar process and ligament of Struthers is rare and only few cases have been described so far ,5,8,9. SApart from compression of the median nerve, the pain may also have a vascular origin. Meda et al. presented a 35-year-old man with compression of the brachial artery and acute ischemia as well as distal embolization caused by a supracondylar process and abnormal high insertion of the pronator teres to the bone spur . The autFracture of the bone spur or concomitant compression of the ulnar nerve has been also described as a rare cause of pain in the presence of supracondylar process . Pedret Surgical excision of the bone spur and release of Struthers' ligament are usually adequate to provide complete and permanent pain relief when conservative treatment fails to alleviate the symptoms ,6. SurgiIn conclusion, a physician should be aware of the presence of supracondylar process in a healthy young child with upper limb pain, numbness, and weakness due to compression of median nerve and/or brachial artery. The symptoms may be bilateral and present simultaneously without a history of trauma or injury. Routine anteroposterior and lateral radiographs are adequate to visualize the pathology and define the diagnosis. Electromyography usually is normal and its value in recognizing the pathology is limited. Surgical excision of the bone spur and its associated Struthers' ligament with subsequent release of the median nerve and/or brachial artery is recommended when conservative treatment could not alleviate the pain and resolve the symptoms. Operative management is associated with prompt pain relief, full functional recovery, and low possibility of recurrence.


Dysuria with lower abdominal pain is a common presentation for a urinary tract infection (UTI), and diagnosis is based on symptoms together with a urinalysis and urine culture suggestive of infection. UTI is uncommon in circumcised males who are not sexually active. When urine culture is negative, alternate diagnoses including, but not limited to, gastroenteritis, severe constipation, appendicitis, or epididymitis need to be considered. In patients with a known urologic history of proximal hypospadias and/or disorders of sexual development, rarer diagnoses also need to be considered. This paper reports the case of a 13-year-old male with a remote history of proximal hypospadias repair, who presented with nonspecific lower urinary tract symptoms. Initially he was treated for UTI. However, urine cultures remained negative despite persistent urinary tract symptoms. On further workup, he was found to have an enlarged and infected prostatic utricle. This report illustrates the importance of considering an enlarged prostatic utricle in the differential diagnoses of patients with chronic lower urinary tract symptoms and a history of hypospadias. Additionally, this case highlights the utility of magnetic resonance imaging (MRI) in clarifying lower urinary tract anatomy in cases where ultrasound is inconclusive. A prostatic utricle is a rudimentary structure in males that is a remnant of the fused caudal ends of the Mullerian ducts. An enlarged prostatic utricle is historically associated with proximal hypospadias and/or disorders of sexual development, although it has been reported in males without any external genital anomalies . The majWe report the case of a 13-year-old male with a history of mixed gonadal dysgenesis and proximal hypospadias repair as an infant who presented with dysuria, abdominal pain, and cloudy urine. Diagnosis was delayed due to the similarity in symptoms with UTI and due to the dilated prostatic utricle being mistaken as a dilated rectum on ultrasound. MRI was ultimately helpful in establishing the correct diagnosis. Physicians should be aware of the association between proximal hypospadias and an enlarged prostatic utricle and its presentation as recurrent UTI. Treatment is indicated for symptomatic prostatic utricles.A 13-year-old male presented to the pediatric emergency department (ED) of a community hospital with lower abdominal pain and pain with urination for 2 days. He had occasional vomiting and loose stools. He had a fever 1 day prior to presentation. In the ED, he had a low-grade fever, a mildly distended abdomen, and lower abdominal tenderness. Urine was turbid with too numerous to count white blood cells (WBCs). He denied sexual activity. His past medical and surgical history was notable for a two-stage proximal hypospadias repair as an infant, which was complicated by the development of a urethrocutaneous fistula. The fistula was subsequently repaired at 3 years of age. He also had a history of mixed gonadal dysgenesis and had a left orchiectomy for a left streak gonad with fallopian tube remnants as an infant. A urine culture was sent, and he was discharged on oral cephalexin with a presumptive diagnosis of UTI.He returned to the ED 2 days later for persistent pain at the end of urination and lower abdominal pain. There were no interval fever or chills, and diarrhea had resolved. He reported worsening suprapubic and right lower quadrant pain, dysuria, urinary urgency and frequency, and foul-smelling urine. He had decreased appetite and activity. Stools had been small and clumped recently, but he denied chronic constipation. His mother reported a small amount of urethral discharge seen on the toilet paper used for wiping after urination. In the ED, he was afebrile and had lower abdominal tenderness on palpation. Genitourinary exam was notable for a circumcised penis, absent left testis, and descended right testis that was mildly tender to palpation but had a normal lie and cremasteric reflex. Urine culture from the prior ED visit showed no growth, although repeat urinalysis at the current visit still showed too numerous to count WBCs. The peripheral WBC count was normal, and C-reactive protein was elevated at 20 mg/dL.Scrotal ultrasound showed a normal right testicle with normal flow and no concerning pathology. Abdominal ultrasound could not visualize the appendix in its entirety, but no secondary signs of appendicitis were seen. Retroperitoneal ultrasound showed normal bladder and kidneys. The radiologist reported circumferential wall thickening of the rectum with an air-fluid level .The differential diagnosis included UTI, appendicitis, scrotal pathology, severe constipation, or gastroenteritis. Scrotal pathology was ruled out by ultrasound. A urine culture had shown no growth, arguing against a bacterial urinary tract infection. Viral UTI was considered unlikely in the absence of significant pyuria. Symptoms of diarrhea, vomiting, and abdominal pain suggested a possible gastroenteritis. The circumferential wall thickening of the rectum with an air-fluid level would make gastroenteritis a reasonable diagnosis. However, the presence of significant pyuria and dysuria would be unusual. The abdominal ultrasound did not fully visualize the appendix. While the absence of secondary signs of appendicitis was reassuring, appendicitis could not be ruled out. To help elucidate any abdominal pathology contributing to his symptoms, MRI of the abdomen and pelvis with contrast was performed. It showed a large, smooth, thick-walled, fluid-filled structure within the pelvis that measured 13.9 × 9.1 × 7.3 cm and appeared to arise from the urethra. The structure was consistent with an enlarged prostatic utricle, which is a Mullerian duct remnant. Comparison with the retroperitoneal ultrasound suggested that the presumed fluid-filled rectum was actually the enlarged prostatic utricle located posterior to the bladder Figures .Further review of the patient's past medical history was revealing. Following penoscrotal hypospadias repair as an infant, he had recurrent UTI. Voiding cystourethrogram (VCUG) was performed and showed no vesicoureteral reflux. However, on the postvoid film, there was a round structure projecting over the lower right pelvis of uncertain etiology. In hindsight, this was likely the prostatic utricle. He developed an urethrocutaneous fistula following his hypospadias repair. At the time of urethrocutaneous fistula repair about 1 year later, a cystourethroscopy was performed, but the neck of the prostatic utricle was not visualized at the time.He was admitted to the pediatric unit and started on IV ceftriaxone. Pediatric urology was consulted, and he was taken to the operating room the following day. Cystourethroscopy was performed and demonstrated debris within the bladder. A small utricular opening was visualized with efflux of debris. The utricular orifice was cannulated, and aspiration yielded 160 mL of thick, mucinous, foul-smelling fluid that was sent for culture. A generous incision of the orifice was performed to allow for adequate drainage of the utricular contents per urethra. He tolerated the procedure well. Culture of the aspirated fluid grew multiple organisms, whereas intraoperative urine culture showed no growth. He was discharged on oral Augmentin. He did well, but symptoms recurred. Therefore, he underwent elective robotic-assisted laparoscopic excision of the prostatic utricle several months later at a tertiary-care center.A prostatic utricle is a rudimentary structure that arises at the level of the verumontanum in the posterior urethra between the openings of the ejaculatory ducts and is present to some extent in all males. Traditional thinking is that the prostatic utricle is a remnant of the fused caudal ends of the Mullerian ducts that regress in males. This view has more recently been challenged by histologic evidence suggesting that the prostatic utricle may derive, at least partially, from the urogenital sinus –3. An enA majority of prostatic utricles may go undiagnosed, as they are asymptomatic when small. When symptoms do occur, they typically present within the first or second decade of life. Symptoms occur if urine and debris are trapped in the pouch causing progressive enlargement with mass effects on the urethra, urinary bladder, or ureterovesical junction. The contents may get infected with bacteria. The most common symptoms of a prostatic utricle are dysuria, urinary retention, epididymitis, hematuria, and urinary incontinence. As these symptoms are suggestive of the more common scenario of urinary tract infection, misdiagnosis is common , 2.There are several case reports of enlarged prostatic utricles in the literature –6. EnlarTreatment of prostatic utricles is recommended only for symptomatic lesions. Treatment of acute symptoms may comprise antimicrobial therapy as well as drainage of cyst contents. Options for drainage include transperineal or transrectal aspiration or cystourethroscopy with catherization and dilation of the utricular orifice. However, these approaches rarely afford definitive treatment. When definitive surgical treatment is indicated, open, laparoscopic, or robotic-assisted laparoscopic excision of the prostatic utricle is performed , 2. Due


Primary benign tumors of the sternum are an exceedingly rare entity. Surgical techniques regarding intervention for these lesions are not clearly defined in the literature given their scarcity. Operative techniques include en-bloc resection of the tumor, and this has proven to be successful in preventing local recurrence despite benign nature of the lesion. Given the often extensive defect created by the excision, reconstruction is frequently necessary; depending on the size of the defect, either autologous bone grafting or the use of synthetic materials may be indicated. This study serves to present two cases of rare primary benign tumors of the sternum, giant cell tumors and osteoma spongiosum and to summarize the available literature. We present a review of the literature of 17sternal giant cell tumor cases reported so far including our patient and unique case of osteoma spongiosum of the sternum, that discusses their surgical management, as well as reconstructive techniques that provided an excellent clinical result and a lack of recurrence on long term follow-up. Benign lesions represent less than 1% of all primary bone tumors, and sternal neoplasms should be considered malignant until proven otherwise.2 The most common types of primary benign sternal tumors are chondromas, bone cysts, and hemangiomas. In this study, we present our experience in the surgical treatment of two extremely rare sternal tumors: giant cell tumors and osteoma spongiosum.Primary chest wall tumors are a very rare entity, with an incidence of 0.04% among newly diagnosed cancers and 5% of all thoracic neoplasms.The medical records of two adult patients with primary chest wall tumors whounderwent resection of the sternum with anterior chest wall reconstruction from October 2015 to May 2020 were retrospectively reviewed. A systematic analysis of electronic databases was performed including the MEDLINE, PubMed, CINAHL, EMBASE and OVID. A total of 11 appropriate full-text case reports and case series in English language were identified through database and reference searches from 1982 to 2021 for giant cell tumor of the sternum with total number of patients of 16 . No sterA 50-year-old female with progressive swelling of the anterior chest wall presented with exertional dyspnea, a dry cough, and persistent retrosternal chest pain developing over the course of a year. Physical examination revealed a round, fixed, non-tender 8×10cm dense mass at the mid-lower aspect of the sternum. The overlying skin was not involved. Laboratory values were normal. A chest computed tomography (CT) revealed a 12.4×9.7 ×8.8cm heterogeneous sternal tumor with anterior mediastinal extension. The mass adhered to the mediastinal vascular structures and pericardium, displacing the heart posteriorly and to the left without signs of invasion . Fine-neThe surgical procedure for management of this lesion began with a midline skin incision over the sternum. Involved cartilages of the 3rd–6th ribs were exposed and excised with wide margin of 2cm from the gross tumor. The sternal resection was performed to macroscopically intact bone tissue (the manubrium was not involved). Internal mammary arteries were identified and bilaterally ligated due to tumor invasion. Wedge resection of the right middle lobe was required due to adherence to the tumor. The resulting defect of the anterior chest wall (10×15cm in size) was covered with a Prolene mesh  , B. StabThe specimen was a 12cm well circumscribed tumor , consistA 42-year-old male presented with uncontrolled, severe, chronic pain in the midsternal region aggravated by minimal activity. Physical examination revealed a 3×4cm area of tenderness and swelling of the upper sternum. Chest CT revealed a cartilaginous mass at the sternomanubrial junction with a widened joint gap, subchondral bone plate with erosive and sclerotic changes, and prominent hyperostosis . AlthougPlanned surgical resection included extirpation of sternomanubrial junction with adequate 1.5cm margins along with bilateral resection of the second and third costal cartilages . The resThe tumor was 1cm in the largest diameter, well circumscribed, consistent with osteoma spongiosum with uninvolved margins . There w3 The multinucleated cells resemble osteoclasts, but are larger and can contain more than 100 nuclei (average 10 to 50). The ratio of cells that form within the same focus may vary. In addition to round-oval mononuclear cells, often there are sites of proliferation of spindle-shaped mononuclear cells with a relatively large number of mitotic figures (up to 500 in 10 fields of view).4Giant cell tumor (GCT) of the bone is an uncommon benign neoplasm of mesenchymal origin. GCT consists of proliferation of multinucleated giant cells in the stroma of spindle-shaped mononuclear cells, which are derivatives of monocytes.5 The term “Giant cell tumor” for its benign origin was suggested in 1919, differentiated from giant cell sarcoma. GCT most commonly occurs in the third and fourth decades of life, with female predominance (1.5:1 ratio); it does not exceed 10% and 20% of all primary and benign bone tumors, respectively. Despite its benign nature, the tumor can invade surrounding structures, and has a high recurrence rate of up to 50%.6 Cases of lung metastasis 1 to 10 years after tumor removal have been described.7 Rarely, less than 2% of malignant GCT, defined as a sarcoma, arise synchronously with GCT, or at the site of a previously resected GCT.8Initially, GCT was described by Cooper in 1818.9 The tumor is classified into three types according to the radiographic appearance: Grade I has a well marginated border and intact cortex; Grade II with a well marginated border without radiopaque rim, the cortex being thin and expanded; Grade III with indistinct borders, cortical destruction, and bulging into the soft tissues. In 1940, Jaffe first classified GCT according to histological characteristics and aggressiveness of the tumor: typical (grade I), aggressive (grade II), and malignant (grade III).10 However, an association has never been shown between the histological grading, tumor behavior, local recurrence rate, and prognosis.There are numerous classification systems for GCT, but the most useful have been introduced by Campanacci in 1987.9 The sternum is an extremely rare location for GCT. Only 16 cases with a morphologically confirmed diagnosis have been described in the literature  localization of the tumor is in the metaphysis and epiphysis of long tubular bones, particularly around the knee, proximal tibia, and distal radius. Less often, the neoplasm affects the scapula, pelvis, vertebral bodies, ribs and hands.terature .6, 7, 1115 analyzed histopathological features of 470 GCTs of bone diagnosed over 20 years. In this review, only one patient (0.21%) had a sternal tumor. This rarity was confirmed in a large series of 568 patients with GCT of the bone at the Mayo Clinic, where a 0.3% incidence was found.22Gupta et al.23According to the literature, the average age of patients with GCT of the sternum is 45 years old (range 28 – 74), which is slightly higher than in patients with GCT of other locations. A differential diagnosis includes more frequent benign  and malignant  neoplasms of the sternum.7 Osteoclast-like cells are not pathognomonic for GCT, as they can be determined in various inflammatory diseases, benign and malignant bone tumors. In the case of a sternal neoplasm, brown tumors of hyperparathyroidism should be excluded by assessing serum parathyroid hormone and calcium levels.21 The immunohistochemical stains for vimentin, S100, pancytokeratin, desmins, and smooth muscle antigen can be useful in uncertain cases.11Fine-needle aspiration is insufficient for the diagnosis of chest wall tumors,and incisional biopsy is required for the accurate morphologic diagnosis of GCT.25 Of note, radiation has no role in the treatment of completely resected tumors.11 To date, there are no generally accepted standards of surgical resection of GCT, and recommendations vary from simple curettage to extensive resection.Surgical resection remains the only radical treatment option for sternal GCT despite successful bisphosphonate therapy and radiotherapy in selected patients with GCT of the bone.21 Futani et al.14 reported extended curettage of the tumor followed by filling of the defect with polymethyl methacrylate (PMMA) with no recurrence observed 7 years after surgery. Similarly, Segawa et al.19 completed the chest wall reconstruction after curettage and cement usage with polypropylene mesh and a titanium mesh plate. This method seems to facilitate recovery, however the curettage has a higher tumor recurrence rate (up to 50%).26 PMMA usage was associated with a recurrence rate of 13%, which is still inappropriately high, and this method is not a treatment of choice in sternal localization of the tumor despite its benign origin. Many surgeons prefer methyl methacrylate (MMA) prosthesis for the anterior chest wall reconstruction; this provides mediastinal protection, elimination of flail chest and paradoxical respiration.21 Some authors used different types of mesh or a polyester patch for the same purpose.21 Engel et al.7 reported on the use of a biomaterial mesh only reconstruction without any respiratory complications. Faria et al.13 reported successful chest wall reconstruction with fascia lata after GCT resection. In our case, we used titanium plates, achieving the desired supportive function.The extent of resection depends on the tumor location, and subtotal sternectomy is the most common procedure.Despite the fact that a giant cell tumor of the sternum is an extremely rare disease, this diagnosis should be in the differential diagnostic algorithm for any neoplasm of the sternum and anterior mediastinum.27 Histologically, the neoplasm can be classified into three different types: compact (eburnated), spongy (mature), and mixed.28Osteoma (osteomata) is a slow-growing, benign, osteoblastic bone tumor that most commonly occurs in the third and fourth decades of life with male predominance (3:2 ratio).29 This type of tumor was first described by Jaffe in 1935 as a small, benign osteoblastic tumor measuring 0.5–2cm in diameter. Morphologically similar lesions larger than 2cm are classified as osteoblastomas. Distinctive radiographic characteristics of OO are a central radiolucency (the nidus), usually less than 1cm, with a perifocal sclerotic zone or cortical thickening, while osteomas exhibit as a radiodense mass.29 Histologically, the central nidus is composed of intersecting trabeculae of osteoid and woven bone, surrounded by rows of activated osteoblasts alternating with osteoclasts within a dilated vascular fibrous stroma.30The entire separate clinical entity is osteoid osteoma (OO), which accounts for approximately 14% of all benign bone tumors, second only to osteochondroma and non-ossifying fibroma. OO most commonly occurs in the first two decades of life with a male predominance (3:1 ratio).30 Only 13 cases have been reported in the literature and included tibia, femur, acetabulum, pubis, ilium, ulna, humerus, clavicle and patella.31 We conducted a focused literature review of PubMed and MEDLINE databases for similar cases of osteomas in the sternum. We identified only 3 previous reports of sternal OO,34 and, to the best of our knowledge, no sternal solitary osteoma cases have been published in the English literature.Lamellar cortical-type bone, resembling normal bone structure, is diagnostic for osteomas. Spongy osteomas are defined as well-differentiated, mature, cancellous bone with intertrabecular hematopoietic bone marrow and minimal osteoblastic-osteoclastic activity. Osteomas are the most common benign lesion of the skull, most frequently located in the paranasal sinuses, while extracranial localization is extremely rare.35 We chose to reconstruct the resultant sternal defect at the sternomanubrial junction with cancellous autologous bone, which is considered to be the “gold standard” bone grafting material, possessing all the necessary basic properties: osteoinduction, osteogenesis and osteoconduction. Autologous bone is histocompatible and nonimmunogenic, thus eliminates the risk of immunoreactions. Dimitriouet al.36 reported an estimated morbidity rate of 19.37% in a total of 6449 harvesting cases. Acute pain and sensory disturbances, including hyperesthesia, dysesthesia or diminished sensitivity are the most common complications (up to 62% of patients) at the donor site. Our patient did not experience any of these postoperative events in short- and long-term follow-up.Surgical resection is the most effective treatment in any type of symptomatic osteoma with a reported success rate of 88 – 100%. En-bloc tumor resection with excision of the surrounding reactive zone reduces the risk of recurrence.This study serves to showcase two rare benign neoplasms of the anterior chest wall that thoracic surgeons may encounter in their practice. Despite the fact that benign chest wall neoplasms are an extremely uncommon entity, GCT and osteoma should be included in the differential diagnosis for any neoplasm of the sternum and anterior mediastinum. With the relative scarcity of literature available to delineate the surgical management of these rare tumors, operative guidelines are not universal. En-bloc resection with wide margins has proven to be a successful approach that has avoided recurrence in long term follow up. Reconstruction is often necessary given the extensive defects created, and should be tackled by either the use of autologous bone grafting or use of synthetic materials. The preferred reconstructive technique is not established. Planning regarding the often complex anterior chest wall reconstruction should be completed utilizing a multidisciplinary approach based on the input from different surgical sub-specialties.