Compound odontoma is a malformation typical of young adults below the age of 20, with a slight preference for the male gender and the anterior region of the maxilla. Clinically asymptomatic, it can be detected during a radiological investigation in connection with the persistence of deciduous dental elements and the impaction of definitive ones. The treatment of choice is excisional surgery and recurrence is a rare event. The need for orthodontic therapy for impacted elements is usually not necessary because in most cases, odontomas are small, circumscribed lesions the size of a permanent tooth. In this article, the diagnostic and therapeutic surgical excision procedure is presented in three patients at developmental age with large compound odontomas associated with at least one retained canine, and in two of the cases, with serious transmigration to the impacted tooth elements. Odontomas are hamartomatous developmental malformations of the dental tissues . AccordiOdontomas are developmental anomalies consequential to the growth of totally differentiated epithelial and mesenchymal cells that generate ameloblasts and odontoblasts . These cTypically asymptomatic, they are revealed on routine radiographs or upon assessing the origin of delayed tooth eruption ,5,6,7,8.Very rarely, the spontaneous eruption can occur with exposition within the oral cavity of the odontoma, and it may be associated with pain, localized inflammation, or infection with suppuration .Odontomas can be diagnosed at any age and in any location of the oral cavity, but more frequently during the second decade of life, on average at 14.8 years . OdontomOdontomas are commonly asymptomatic and constitute casual findings. Clinical signs may be delayed eruption and persistence of the deciduous teeth. In severe cases, infection or regional lymphadenopathy may be observed .Management usually consists of surgery to prevent further complications with the permanent tooth eruption in the paediatric population ,10, and Following surgical removal of the odontoma, orthodontic therapy is often not prescribed to reposition any impacted tooth elements . In the This article presents three clinical cases of patients at developmental age diagnosed with large compound odontomas, associated with the inclusion of at least one permanent canine and impacted teeth transmigration in two of the cases. The diagnostic process and surgical treatment of choice are described, in which the two transmigration cases led not only to the removal of the odontoma but also to the extraction of the impacted and transmigrated tooth elements. In all cases, histopathological diagnosis was performed and was compatible with compound odontoma.Besides, literature searches of free text and MeSH terms were performed using PubMed and Google Scholar from 2000 to 2022. All searches were conducted using a combination of subject headings and free-text terms. The keywords used in the search strategy were as follows:(“compound odontoma” AND “complex odontoma”) AND  AND (“tooth impaction” OR “impacted teeth” OR “impacted tooth” AND “tooth transmigration”).The aim of this review was to hightlight the anatomical, demographic, gender and dental parameters associated with complex and compound odontoma.Inclusion criteria were as follows: (a) prospective studies; (b) retrospective studies; (c) case series; (d) in-vivo studies; (e) studies published in English, French, Polish and Albanian. These criteria have been broadly selected to be as sensitive as possible. The exclusion criteria were as follows: (a) in-vitro studies; (b) articles without statistical analysis; (c) abstracts and letters to the editor.A manual search was also conducted to try to find other additional studies as well. Based on the inclusion criteria, two authors (MM and GDG) reviewed the titles and abstracts and selected the studies from the literature independently. The full text of each study was then read to decide whether it could be included or not. Disagreements in this case were set on by consensus between both authors or by discussion with another author (AN).A 14-year-old female patient, treated in our paediatric surgery unit in 2021 .The second clinical case concerns a 14-year-old male patient who was referred for consultation to our paediatric oral surgery unit. The patient was operated on in November 2021 . The diaThe third case presented here is that of a 15-year-old male patient. The surgery was performed in July 2021 .The search strategy identified 124 potential articles: 33 from PubMed and 91 from Google Scholar. After removal of duplicates, 91 articles were analysed. Subsequently, 71 papers were excluded because they did not meet the inclusion criteria and were not relevant to the subject of the study. The remaining 20 papers were included ,30,31,32The studies included  in the pCompound odontoma is a benign odontogenic tumour and it is usually diagnosed in young adults during regular radiological examination performed to assess the reason of a missing or mispositioned tooth in permanent dentition .At the clinical level, compound odontoma can often be associated with anterior teeth misalignment and tooth eruption disorders, with possible impaction and delayed tooth eruption. One quarter of patients are asymptomatic, but compound odontoma can also be characterized by pain (13.3%) and swelling (8.9%) . The preSurgical removal is the usual treatment and recurrence is rare . In the clinical cases presented here, it was possible to refer the patient for orthodontic treatment of the impacted canine only in the first case, when the inclusion concerned the upper left canine. In this case, the odontoma blocked the physiological eruption of the canine, but did not cause it to be misaligned. In fact, the canine position was vertical, even if slightly vestibular, placed above the odontoma.In the remaining two cases, the position of the odontoma was in the front part of the mandible and was associated with the inclusion of the canine, which was unfavourable for orthodontic treatment. In fact, in both cases, we proceeded with both the surgical enucleation of the odontoma and the extraction of the impacted canine, and in one case, also the extraction of the lateral impacted transmigrated incisor. In both cases, the large volume occupied by the odontoma, and late diagnosis certainly contributed to the displacement of the canine from its physiological site towards the mesial position, almost completely horizontal towards the bottom of the mandible on the inferior cortex, in line with the entire symphysis area of the chin. This type of tooth displacement is called transmigration. Transmigration most commonly concerns the permanent canines and is characterized by the movement of a non-erupted tooth that crosses the median line of the mandible and goes to position itself in the opposite part of the anterior mandible, as in cases 2 and 3 of this report . TransmiThe presence of cysts and tumours can alter the eruption pathway and cause tooth transmigration. The transmigrated teeth typically present unilaterally with a certain degree of angulation. In case 2, the angulation was almost horizontal to the lower jawline, while the transmigrated canine in case 3 presented a 60-degree angulation to the corresponding long axis of the first lower premolar ,39.The treatment of choice for transmigrated canines in association with vast compound odontoma is extraction. Orthodontic reposition of transmigrated canines is usually reserved for mild cases .Few cases in the literature could be found with transmigrated canines being transplanted following endodontic therapy .However, the management of bone gaps after surgical excision treatment of large lesions may require regenerative approaches using a combination of three-dimensional structures and new therapeutic means .This article has documented three cases of compound odontomas: one in the upper premaxilla and two in the mandible in patients at developmental age. All the cases were associated with canine impaction; however, only the maxillary-impacted canine underwent orthodontic treatment after surgery. The two mandibular canines associated with compound odontoma were transmigrated and no orthodontic therapy was possible due to the challenging near-horizontal position at the lower portion of the anterior mandibula.Although the radiological and macroscopic features following surgical removal are pathognomonic, histological examination is always performed. As a diagnostic investigation in preparation for surgery, CBCT offers details on the morphology of the odontoma, its relationship with the surrounding dental structures, and the cortical bone profiles of the affected jaws.


Prostate cancer is one of the most common cancers that metastasize to the bone. Bone metastasis is usually osteoblastic, and diffuse osteolytic lesion on presentation is unusual. Here, we are reporting MRI image of patient presented with diffuse osteolytic lesion and found to have metastatic prostate cancer. Prostate cancer can present as diffuse osteolytic bony lesions, and clinician should keep in mind such differential when encountering such presentation. It is not related to movement and is present both day and night. The patient reported 8 kg unintentional weight loss during the last 6 months. The patient reported some leg weakness also during the last few days. He denied fever and sensory symptoms with no urinary or fecal incontinence. He denied lower urinary tract symptoms. The patient is a nonsmoker and drinks alcohol on social occasions. He has no family history of cancer. The examination was unremarkable except for mild lower limb weakness due to pain in the back and lower limbs. Digital rectal examination showed flat prostate, which is hard in consistency. Laboratory workups were shown a normal complete blood count and renal function test. His serum alkaline phosphatase was 153 U/L, and serum prostate‐specific antigen was 633 ng/mL. MRI spine (FIGURE 1)revealed widespread multiple variable size osteolytic metastatic lesions in the cervical, dorsal, lumbar, and sacral vertebral bodies and a few appendicular sites showing T1 low, T2/STIR bright signal with postcontrast enhancement mainly at C5, C7, T2, T3, T6, T7, T9, T11, T12, and to, L4, S1, and S2 levels. Computed tomography of the abdomen and pelvis redemonstrated multiple spines and diffuse destructive lytic pelvic bony lesions. It showed large expansile soft tissue lesions with bony destruction involving bilateral sacroiliac joints, multiple enlarged iliac lymph nodes, and slightly enlarged non‐homogenous prostate. PET scan confirmed our findings. CT‐guided right iliac bone lesion biopsy was done, and histopathology showed metastatic adenocarcinoma of prostatic origin. IHC stains for NKX3.1 and AMACR showed that tumor cells are robust and diffusely positive and IHC stain for PSA is focally positive. The patient was diagnosed with metastatic prostate cancer, and we planned to start him on androgen deprivation therapy and antiresorptive therapy.2Prostate cancer is one of the most common cancers that manifest as bone metastasis. The nature of the bone lesion in prostate cancer is typically osteoblastic, but osteolytic bony metastasis as first presentation has been reported.Abdulrahman Al‐Mashdali: Writing – review and editing. Noor Jasim: Data curation; visualization. Mohamed Salem Ismail Khalil: Supervision.The authors have no conflict of interest to declare.Written informed consent was obtained from the patient for the publication of this case report.


Penile fracture is defined as a tear of tunica albuginea that covers the corpus cavernosum during an erection. It is a rare finding that both the corpora cavernosum and corpora spongiosum are involved in penile fracture. Herewith, we reported a rare case of 44 years old presented with penile fracture during woman on top sex position with both corpora cavernosum and corpus spongiosum rupture with urethral disruption. On clinical examination, the penis was swollen, and there was a sudden loss of erection and ecchymosis. Cystoscopy examination revealed urethral rupture. Emergent surgical repair was then performed. During emergency surgery, we found a defect of 3 cm in bicorporal cavernosa with urethral and corpus spongiosum disruption. The penis was degloved, and debridement with water-tight suturing of tunica albuginea was performed to repair the tear in corpora cavernosa. End-to-end anastomosis urethroplasty with spatulation was also performed to repair the urethra. After 21 days following surgery, erectile function was good and no difficulties in voiding function as shown in uroflowmetry result with Qmax >15 mL/s. The patient had a favorable recovery. This was a rare case report, and with early and prompt surgical intervention, this case could result in a good outcome in preserving erectile function and voiding function. •Penile fracture is an emergency condition in field of urology•The presence of tricorporal fracture involving both corpora cavernosa and corpora spongiosum was considered rare case•The diagnosis of penile fracture was based on clinical presentation of history taking and physical examination•Immediate surgical treatment involving penile degloving and surgical repair resulted in promising outcome The penis was swollen after the patient had vigorous sexual intercourse with his wife so-called “woman on top” sex position. There was a crack sound followed by swelling afterward. He suddenly felt severe pain, followed by immediate loss of erection. Blood was discharged from the tip of his penis. There was no previous history of diabetes mellitus, hypertension, or previous history of surgery. He denied any sexual disorder and history of PDE-5 inhibitor use. On physical examination, his penis was swollen showing the signs of ecchymosis . UrethraThe patient was immediately scheduled for an operative procedure. Suspected urethral disruption with its localization was evaluated prior to surgical repair using cystoscopy, and eventually revealed urethral rupture A. An incAn uneventful post-operative period following surgery was found. After 21 days of the follow-up period, uroflowmetry and urethrography examination revealed a normal result. Erectile function was normal with a normal erection hardness score of 4. No deformity in penile curvature was seen, and no difficulty in voiding function was revealed, as shown in the uroflowmetry result with Qmax >15 mL/s. The patient did not complain of any pain during sexual intercourse. 3This is a rare case of penile fracture involving the tricorpora which are both corpora cavernosa, and corpus spongiosum with urethral disruption. This case report demonstrated appropriate management with a rare presentation of penile fracture involving the tricorpora. The penis is consisted of erectile tissue, which was arranged in columnar order. There were two dorsolateral corpora cavernosa and one ventral corpora spongiosum, each was coated by the tunica albuginea. The urethra runs through along the corpora spongiosum and forms the glans penis at its tip. The ventral corpora spongiosum was coated by Buck's fascia The diagnosis of penile fracture was based on history taking and physical examination, in which 90 % of the symptoms were sudden cracking or snapping sound, immediate pain, and a significant subcutaneous hematoma Retrograde urethrography (RUG) should be considered in this case because it is inexpensive, easy to perform, and highly accurate Compared to conservative treatment, surgical treatment has produced better outcome and shorter hospital stays with less complication rate. Surgical intervention with the closure of the tunica albuginea can ensure the lowest rate of negative long-term sequelae and has a favorable effect on the psychological wellbeing of the patient. A previous study reported the comparison of surgical and conservative management with 50 % and 4 %, respectively The importance of penile degloving, in this case, was for a comprehensive search to check any missed injuries 4Penile fracture is one of the emergency conditions in urology. Bilateral rupture of corpora cavernosa with urethral disruption is an uncommon case. The diagnosis of penile fracture was based on clinical presentation. The classic symptoms were snapping or cracking sounds, followed by immediate detumescence and severe pain. Prompt surgical treatment involving penile degloving and surgical repair resulted in a promising outcome.Ethical approval has been acquired in this study.No funding was granted in this study.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.Conceptualization – GWK, MY, IAR; Data curation – GWK, MY; STJ; Materials – GWK, MY; Formal Analysis – GWK, MY, IAR, STJ; Investigation – GWK, MY, IAR; Methodology – MY, IAR; Supervision – GWK; Writing original draft – GWK, MY, IAR; Writing, review and editing – GWK, STJ.Not applicable.Gede Wirya Kusuma Duarsa.No conflict of interest in this study.


Fetus in fetu (FIF) is a rare congenital disorder in which a deformed fetus forms inside a normal one, mostly within the abdominal cavity. In this case study, we present a triplet pregnancy where one fetus was formed within a viable fetus. This was noted in prenatal imaging and upon delivery. Radiological investigations were conducted, including plain X-rays, abdominal computed tomography, and magnetic resonance imaging, and a preliminary case of FIF was diagnosed. Following this, surgical resection was planned and done soon after birth with histopathology confirmation of diagnosis, and it revealed no evidence of somatic malignancy. Currently, the patient is in stable condition and is being followed up with serial ultrasound imaging with alpha-fetoprotein levels to detect recurrence. Fetus in fetu (FIF) is a rare congenital malformation with less than 200 cases reported worldwide and an incidence of one per 500,000 births . It invoThis condition has been described in both pediatric and adult populations; however, it appears more commonly during early childhood with a 2:1 male-to-female ratio [In this report, we present a rare case of FIF in a four-month-old female infant, who was a product of an in vitro fertilization (IVF) twin pregnancy to a primigravida mother. To the best of our knowledge, this is the first case of FIF to be reported within our institution as well as in our country of practice, Bahrain.In this case, we report a four-month-old female infant who was the product of an elective lower segment C-section (LSCS) due to twin pregnancy with a breech presentation of the first twin. The pregnancy was conceived through IVF by a 30-year-old primigravida woman. During her pregnancy, an antenatal abdominal ultrasound demonstrated an abdominal cystic lesion with ascites in one fetus.Our twin of interest, in this case, had an APGAR  score of 9, 10, and 10 upon birth. Noticeably, following her first postnatal examination, she appeared normal without any dysmorphic features. However, her abdomen appeared to be distended on inspection. Further focused physical examination showed a large abdominal mass in the left hypochondrium crossing the midline. Following this, she was admitted into the neonatal intensive care unit (NICU) for further investigations. The neonate was kept nil per oral (NPO) and kept on fluid maintenance until the workup was completed. Initially, plain abdominal radiographs demonstrated a large cystic shadow in the left hypochondrium with radio-opaque shadows, as seen in Figure Moreover, a computed tomography (CT) scan of the abdomen and pelvis and a magnetic resonance imaging (MRI) scan were done, which showed a large complex mass occupying most of the peritoneal cavity, mainly located on the left side. Images showed that the mass contained fat, fluid, soft tissues, and parts of the fetal skeleton and limbs, along with neural tissues, with the mass having an arterial supply from the aorta. These findings can be seen in Figures Furthermore, a three-dimensional (3D) reconstruction of the bony skeleton was done, demonstrating the fetal skeletal bones and limbs present within the abdominal cavity of the infant, as seen in Figure Consequently, the surgical team performed a laparotomy on the infant to resect the mass along with its capsule. Upon gross examination following resection, the mass had a well-defined capsule and multiple large vessels feeding it, which corresponded to the imaging reports. It weighed 366 grams, and its contents were bone, hair, and fetal limb remnants, all of which were further taken for histopathology. The histopathology report showed findings consistent with FIF malformation with no evidence of somatic malignancy.The patient’s postoperative period was uneventful, in which she was extubated and discharged in good condition. Concurrently, preoperative tumor markers were sent, but the sample was insufficient, and no readings were available. One week after the operation, alpha-fetoprotein (α-FP) was repeated, and the result came elevated, measuring 36964.4 micrograms/L. However, follow-up α-FP levels showed regression, with the latest level measuring around 14136.5 micrograms/L at six months postoperatively. A follow-up abdominal ultrasound was done one-year postoperatively and showed no significant abnormal soft tissue at the site of the surgery.When discussing FIF, multiple theories arise regarding the pathogenesis of this rare condition. One theory is the uneven division of the developing blastocysts’ totipotent cells, resulting in the encasement of the smaller product of the division into the larger maturing embryo . This leTypically, FIF is an intra-abdominal pathology, although some cases have reported this pathology being located within the cranial cavity or even the scrotum . The iniAdditionally, a contrast-enhanced CT scan can help to further characterize the mass and can be of great value in assessing the surrounding structures and vascularity for planning the surgical resection. The typical CT findings of FIF include a rounded or tubular mass with a fat component surrounding the vertebral bony axis . MoreoveMRI is currently being used more frequently to diagnose this entity due to its ability to provide detailed tissue characterization. Further, its lack of ionizing radiation makes MRI a safe tool to confirm the diagnosis in the antenatal phase and during follow-up in the postnatal period. It can provide a thorough evaluation of the bony structures, including the vertebral bodies, rib-like and long bones, limb buds with primitive phalanges, and the cranial fossa and orbital sockets.Although not as common as a CT or MRI scan, reconstructive tools such as multiplanar reformation (MPR) and volume rendering technique (VRT) can provide 3D images and aid in the evaluation of the surrounding anatomical structures and the exact location of the lesion, as was done in our case. FIF’s diagnosis primarily depends on radiological imaging and a definitive diagnosis can be made by histopathology following resection, markers such as α-FP levels can also help in this condition’s diagnosis and follow-up . DespiteThe treatment of surgical resection of the encapsulated fetus relieves the obstruction and prevents further compression. Complete surgical resection of the surrounding sac is important since leaving behind a portion of the mass capsule may facilitate recurrence, although a case by Magnus et al. in 1999 reported that it recurred as a yolk sac tumor four months after the operation [When comparing the management of this case with that of other FIF cases worldwide, we notice that the approach is similar to the one undertaken in this case. Most authors report that initial suspicion of the diagnosis begins with antenatal ultrasound within the final trimester of the pregnancy and Mills et al. described the importance of antenatal ultrasound in the diagnosis of FIF -9. This Further investigations usually began with plain radiographs followed by further imaging in the form of a CT scan or an MRI depending on the location of the FIF. Once the preliminary diagnosis was done, complete surgical resection was undertaken during infancy. This was seen in the cases reported by Magnus et al. and Nanjaraj et al., in which the approach was quite identical to that of our case even though the locations of the FIF were intrahepatic and intracranial, respectively ,9.Finally, frequent postoperative surveillance is recommended, especially if the sac could not be completely resected. This may be achieved with moderate sensitivity using serum tumor marker concentrations and imaging, preferably modalities without ionizing radiation, such as ultrasound or MRI.FIF is a rare congenital condition that should be a part of the differential of infants presenting with pelvic-abdominal mass. It has similar imaging and histological features as teratoma; however, the presence of axial and long bones within the mass in radiological imaging is diagnostic of the entity. The ideal imaging modalities to diagnose and plan for surgical intervention are either a CT scan or an MRI. However, given the risks of radiation exposure, MRI is more frequently used both pre- and postnatally.


Rosai-Dorfman disease  is a rare reactive histiocytic disease, classically involving the lymph nodes of the neck, but it can also occur in extranodal sites. Isolated spinal involvement is rare but important to identify as it can mimic malignancy with males being affected more than females. We present a case of a 52-year-old female patient who had breast cancer and was admitted with long-standing anal and sacral pain. The MRI lumbar spine showed a mass concerning for metastasis. CT-guided biopsy results showed sheets of histiocyte-like cells, some of which were positive with S100 and showed emperipolesis along with plasma cells, lymphocytes, and neutrophils. Features were in keeping with Rosai-Dorfman syndrome. The patient responded to steroids. The familiarity with this entity saved the patient from going through the agony of this being considered a metastasis of her primary malignancy. Rosai-Dorfman disease (RDD) was first described in 1969 by Rosai and Dorfman, the etiology of which is mostly unknown . DiagnosA 52-year-old female with left breast carcinoma presented with long-standing anal and sacral pain. The patient was alert and oriented. There were no cerebellar or meningeal signs. She had a completely normal physical/system examination. MRI Spine showed a multilobulated mass lesion located in the sacral spinal canal at the level of S1 and S2 causing extrinsic bony erosions in the posterior aspect of the body of S1 and S2 with the posterior extra spinal extension of the lesion through the posterior arch of the S1 and S2 vertebrae. The proximal part of the lesion appeared to be intradural in location and the distal part of the lesion appears intradural and extramedullary in location. The lesion showed intermediate signal intensity on T1 weighted images and bright signal intensity on short tau inversion recovery (STIR) sequences with enhancement on post-contrast images /CT using 18F-FDG can also be used to diagnose RDD and show numerous enlargements, as described by Dhull et al. . HistopaIsolated RDD chancing on its own is a very rare histiocytic disease along with it being an exceptionally difficult diagnosis. The diagnosis is mainly dependent on histological and immunophenotypical testing. Its radiologic features are similar to other dural-based lesions. Disease progression is extremely rare. Surgical treatment relieves the compressive symptoms and remains the treatment of choice. Neoadjuvant therapies, steroids, and other treatments are available, although further extensive systematic investigations should be made in this field.


Stump appendicitis is a rare, delayed complication of appendectomy. It is seen following both open and laparoscopic appendectomy and may occur weeks to years following the initial appendectomy. We report two cases of stump appendicitis seen at our hospital. Both cases were diagnosed based on radiological findings and successfully managed conservatively with antibiotics. Although the usually recommended treatment for stump appendicitis is completion appendectomy, conservative management may be suitable for some patients. This report highlights the possibility of utilizing a conservative approach in the management of stump appendicitis compared to the recommended operative intervention. Awareness of the possibility of stump appendicitis is crucial for early diagnosis and treatment, to prevent potentially catastrophic complications. However, this diagnosis is often not considered in patients with a prior history of appendectomy.2 Failure to identify this condition may lead to increased morbidity and hospital stay as well as complications such as perforation, abscess formation, and sepsis.Stump appendicitis is a rare, long-term complication of appendectomy with a reported incidence of 1 in 50,000.Here, we report two cases of stump appendicitis that presented to our emergency department with abdominal pain, varying durations following an appendectomy. Both patients were treated conservatively with antibiotics and made an uneventful recovery.A 41-year-old gentleman presented to the emergency department with abdominal pain in the right lower quadrant for 3 days. There was no history of fever, vomiting, altered bowel habits or urinary symptoms. He had undergone an uneventful appendectomy 2 years ago at our centre after presenting with similar symptoms. Family history and socioeconomic history were unremarkable. On admission, he was afebrile and his vitals were stable. Abdominal examination revealed tenderness in the right iliac fossa. His laboratory tests were normal.As a part of routine investigation abdominal ultrasonography (USG) was advised which revealed an appendicular stump with surrounding periappendiceal fluid collection. An abdominal Computed Tomography (CT) scan revealed inflammatory changes in the region of the cecum with adjacent fat stranding and lymphadenopathy -3. HenceThe patient was admitted to the general surgery ward and started on IV (Intravenous) antibiotics (cefoperazone one gram two times a day and metronidazole 500 mg three times a day for 5 days). He was kept nil per oral for 5 days and was managed with analgesics and antiemetics as well. He made an uneventful recovery and was discharged after 6 days with 1 week of oral antibiotics . He did not have any difficulties at 2 weeks follow-up. Follow-up USG reports showed resolution of the inflamed stump.A 23-year-old lady with a previous history of laparoscopic appendectomy 1 month back, presented to our emergency department after 2 days of rightsided abdominal pain, fever, nausea, and vomiting. The pain was initially dull aching in character, worsened gradually and was associated with decreased appetite. She denied having altered bowel habits or urinary symptoms. Her last menstrual period was 10 days before the presentation. On physical examination, vitals were stable. On abdominal examination, tenderness was present in the right iliac fossa. Rebound tenderness was present. Inflammatory markers were not raised. As a part of routine investigation, abdominal ultrasonography (USG) was advised which showed a small stump appendix with surrounding inflammatory changes and fluid collection. Abdominal CT was done which supported the USG findings -5.A stump of the appendix measuring 8 mm with an air dock within the lumen was visualised along with inflammatory changes. A conservative approach with IV antibiotics was prescribed similar to case 1. Supportive treatment was given and the patient improved remarkably. She was discharged after a 7 day course of antibiotic therapy with oral antibiotics similar to case 1. She remained asymptotic for 1 month following discharge.3 Rose, in 1945, was the first to describe stump appendicitis in two patients who had previously undergone appendectomy for acute appendicitis.4 It is a rare occurrence with a reported incidence of 1 in 50000 cases but the real incidence is expected to be much higher as this condition is often underestimated.4 The incidence has been increasing recently which may be attributed to increased awareness among treating physicians as well as the easy availability of CT scans.Stump appendicitis is defined as the interval repeated inflammation of remaining residual appendiceal tissue after an appendectomy, either due to impaction of a fecolith or secondary to an ischemic process.5 Lack of a three-dimensional perspective and the absence of tactile feedback may obscure the base of the appendix, leaving behind a longer appendicular stump which may act as a reservoir for fecoliths and predispose to the development of stump appendicitis. Although a majority of reported cases have occurred following an open appendectomy, it may not accurately reflect the incidence in comparison to the laparoscopic technique, as the latter is relatively newer to the traditionally practised open method.5The laparoscopic technique has been implicated as a factor in the recent increase in the incidence of stump appendicitis.6 In our cases, stump appendicitis developed just 1 month following a laparoscopic appendectomy in contrast to 2 years after an open appendectomy.The time interval between initial operation and recurrence of symptoms could range from 2 weeks to years after appendectomy. It has been suggested that this duration may be significantly shorter following laparoscopic appendectomy.7 These nonspecific symptoms along with a past history of appendectomy make the diagnosis challenging. Hence, radiological examinations are of great significance in diagnosing this uncommon phenomenon. An abdominal CT scan is considered the gold standard and shows findings similar to acute appendicitis including pericecal inflammation, cecal wall thickening, free or loculated fluid in the right paracolic gutter, and infiltration of the surrounding fat. A long appendiceal stump might be visualised as a tubular, thick-walled, fluid-filled, enhancing structure.8 In both our cases, simple abdominal ultrasound was able to suspect the condition accurately and was further strengthened by typical CT scan findings.Clinical presentation of stump appendicitis is similar to acute appendicitis. Common symptoms include abdominal pain, usually in the right iliac fossa associated with nausea and vomiting.9 More extensive surgery such as ileocecectomy and right hemicolectomy may be required in some cases. Conservative treatment with antibiotics is proving to be effective in some instances, especially in the absence of appendicoliths or evidence of perforation, as seen in our cases.10 Careful follow-up is mandatory in these cases to prevent the recurrence of chronic appendicitis.Completion appendectomy, either by laparotomy or laparoscopy has been considered the treatment of choice and is generally sufficient when the base of the appendix can be identified and the cecum is not significantly inflamed.Stump appendicitis is a rare but serious complication of appendectomy, whose incidence has been increasing in recent years. As it presents with vague, nonspecific symptoms, a high index of suspicion is required for diagnosing the condition. Imaging techniques like USG or CT scan may aid in establishing the diagnosis. Although stump resection is the preferred treatment in most reported cases, conservative management with antibiotics may be an effective, alternative treatment option.


Persistent hyperplastic primary vitreous (PHPV) in a patient with tuberous sclerosis (TS) has been described in one previous case report in 1999. Otherwise, there is no literature around this potential association. We describe a case of an infant with TS and PHPV.An 11-month old male was under investigation for leukocoria, microphthalmia and suspected PHPV after being seen in ophthalmology clinic. He presented to hospital with seizures and was diagnosed with TS on imaging. Imaging also showed the known microphthalmia and a mass associated with the lens. Subsequent paediatric ophthalmology review and examination under anaesthesia confirmed microphthalmia, PHPV and a retrolental mass which was thought to represent total retinal detachment or a retinal hamartoma within a retinal detachment.This is the second case report of PHPV in a patient with TS. The previous case report postulated that the atypical location of the retinal hamartoma was secondary to the abnormal globe development in PHPV. Tuberous sclerosis (TS) is a rare neurocutaneous disorder characterised by hamartomas of multiple organs, including the brain, kidney, heart, skin and eyes. It is inherited in an autosomal dominant pattern, though two-thirds of cases are de novo mutations . The repAn 11-month old male was referred to ophthalmology for leukocoria and found to have unilateral microphthalmia and suspected PHPV on ultrasound, with a subsequent examination under anaesthesia planned. One month later, he presented to the emergency department with a seizure in the context of a coryzal illness. He was treated with antiepileptics and antibiotics and proceeded to have a CT head and MRI brain which showed findings consistent with tuberous sclerosis; multiple subependymal nodules in the lateral ventricles, some calcified, and diffuse subcortical low attenuation suggestive of tubers Fig. . The masFollowing discharge, outpatient paediatric ophthalmology and examination under anaesthesia confirmed unilateral right globe microphthalmia with cataract and PHPV. The retrolental mass was thought to represent total retinal detachment or a retinal hamartoma within a retinal detachment. The left eye was normal. No treatment was offered due to the poor prognosis of the right eye and the patient continues to have ongoing ophthalmology follow up. The family is awaiting genetics consultation. Informed consent for this report was gained from the patient’s parents.This case report describes a patient with TS with a rare ophthalmic finding of microphthalmia, PHPV and retinal detachment, with the possibility of a retinal hamartoma within a retinal detachment. A similar finding has been described in a case report in 1999 in Canada . A 6-weePHPV is a rare congenital developmental malformation, caused by the failure of regression of the primary vitreous . It usuaAside from the aforementioned historical case report, there is no known association between TS and PHPV. Recently, genetic studies have found that microphthalmia-associated transcription factor (MITF) transcription and expression is reduced by mutations in TS genes, TSC1 and TSC 2 . MITF muA strength of this case is that despite the rare occurrence of the conditions, we were able to make these diagnoses under the care of subspecialist paediatric radiologists and ophthalmologists. However, limitations include the absence of histopathological confirmation and genetic analysis currently.We believe this is the second published case report of a patient with TS and PHPV, resulting in either total retinal detachment or a retinal hamartoma within a retinal detachment.


Though the majority of differentiated thyroid cancer (DTC) patients have a good prognosis after careful and standardized therapy, approximately 13% to 15% of DTC cases show surprisingly aggressive behavior and invasion of the surrounding structures, and a few progress to unresectable diseases. In this study, we report a case of an inoperable locally advanced DTC patient who underwent a curative operation after treatment of preoperative monotherapy of apatinib in a short time.A 64-year-old woman complained of dysphagia due to large cervical mass, which severely invaded the left esophagus at the junction of the neck and thorax.The female patient was diagnosed with locally advanced papillary thyroid cancer (PTC) by cytopathology and it was difficult to perform a safe and complete removal.Apatinib  was initially used to treat this patient as a neoadjuvant therapy.131I ablation therapy were performed. At the 1-year follow-up evaluation, no tumor recurrence or metastasis was observed.Six weeks later, the tumor dramatically shrunk from 56 × 37 mm to 29 × 26 mm with well-controlled mild hypertension. After a 10-day interval of apatinib withdrawal, complete tumor excision was accomplished through cervical incision without esophageal fistula. Postoperative thyroid stimulating hormone suppression and radioiodine Preoperative short term targeted treatment with apatinib for locally advanced inoperable DTC may become a promising neoadjuvant therapy that, can reduce the tumor size and decrease stage, thus making the complete and safe removal of the lesion feasible. DTC comprises the vast majority (>90%) of all TC, and generally carries good prognoses after standard surgery with thyroid stimulating hormone (TSH) endocrine inhibition and sometimes also with radioactive iodine (RAI) therapy. Nevertheless, approximately 13% to 15% of DTC cases could show surprisingly aggressive behavior and involve the surrounding structures, such as the recurrent laryngeal nerve (RLN), trachea, esophagus, larynx, pharynx, or major cervical vessels, which complicates the surgical treatment and dramatically increases postoperative complications and mortality or even leads to tumor progression to unresectable disease.Thyroid cancer (TC) is the most common endocrine malignancy and caused 41,000 deaths worldwide in 2018. Several kinds of methods can be applied, such as external beam radiation therapy (EBRT), systemic chemotherapy (ChT), molecularly targeted treatment, radioactive 125I seed implantation, chemoembolization, thermal (radiofrequency or cryo-) ablation, and ethanol ablation, but complete and adequate surgery for TC represents the main-stay of the multimodal treatment approach and the most significant determinant of outcome. Nevertheless, the surgical management of advanced TC is complex and challenging, and in some cases, it is difficult or impossible to perform an effective surgery. In that specific context, how can we improve the clinical stage and create an opportunity for surgery? Similar to breast-conserving surgery or anus-preserving operation combined with preoperative chemotherapy for the treatment of middle-advanced cancer, neoadjuvant therapy would be worthwhile to try in DTC.In fact, few guidelines are dedicated to the optimal approach of these locally advanced TCs. Vascular endothelial growth factor (VEGF) and its receptor are considered potent factors in angiogenesis and are excessively expressed in solid tumors; thus, they have been confirmed to be promising targets for the treatment of neoplasms. Apatinib is a novel, oral, small-molecule tyrosine kinase inhibitor (TKI) that highly and selectively targets vascular endothelial growth factor receptor 2 (VEGFR-2). Recent clinical trials have revealed its hopeful anticancer effect in various solid tumors,,8 and a similar result in TC was found in our team's previous research. Herein, we report a case of a locally advanced inoperable DTC patient who underwent complete safe removal after undergoing preoperative short term apatinib targeted treatment, with little drug-related side effects and almost no postoperative complications.Pathological angiogenesis is an important characteristic and essential for tumor growth, invasion, and metastasis, especially for solid tumors.2The study was approved by the Ethics Committee of Shanghai Jiao Tong University Affiliated Sixth People's Hospital. Written informed consent was obtained from the participant. A 64-year-old woman was admitted to our hospital, who complained of gradually worsening dysphagia for half a year and could currently only intake fluids. This patient was diagnosed with PTC by cytopathology at the local hospital and it was difficult to perform total surgical resection due to the severe tumor invasion of the esophagus in the cervicothoracic junction. She had no history of high blood pressure, diabetes, hyperthyroidism, autoimmune diseases, or family illness. After physical examination, we found a large hard and immobile mass located in the left neck (7 × 6 cm in size). The hormone levels of thyroid function were normal except for a significant increase in thyroglobulin (Tg) .A repeat ultrasound inspection revealed a 36 × 39 × 73 mm hypoechoic mass with irregular modality, an ill-defined border, heterogeneous internal echoes, and an abundant color flow signal in the left neck. Moreover, there was no enlarged cervical or supraclavicular lymphadenopathy. Ultrasound-guided core biopsy in the left lobe of the thyroid gland was performed, and the pathology indicated classic PTC Fig. . A contrThe multi-disciplinary team (MDT) suggested that it was difficult to resect the tumor completely and safely due to the severe involvement of the surrounding structures of the trachea and esophagus. It might become a palliative operation if surgical treatment is selected as the primary choice, and the risk of esophageal fistula could also be high. Considering the potential antineoplastic activity and rapid response of apatinib in several kinds of malignancies, our team proposed off-label preoperative targeted therapy for this specific case. Of course, economic factors cannot be ignored: the cost of apatinib is only one-half that of sorafenib, and apatinib is also more available than sorafenib in China. Treatment with 500 mg apatinib orally once per day was started on February 14, 2019. Fortunately, the only adverse drug reaction that occurred was mild hypertension. One month after therapy, the patient benefited from favorable clinical tolerability and safety, and an interval CT scan indicated tumor shrinkage to 36 × 25 mm in size and a significant reduction in the degree of pressure on the esophagus  during the first month of follow-up. The level of TSH was adjusted to <0.1 mU/L by levothyroxine dosing to reduce the risk of disease recurrence. Six months later, neck ultrasonography found no residual TC or normal thyroid tissue, chest CT scan and whole-body bone scan Fig.  indicate3 Extensive gETE portends an increased incidence of local recurrence and regional and distant metastasis. Though management is controversial, it is certain that the most significant approach is to remove the neoplasm en bloc to achieve local control and extend disease-free survival. However, the balance between radical surgery and the functional preservation of the anatomical structures involved must be taken into account. There is no doubt, that surgical therapy for locally advanced DTC is challenging, and the resection scope remains controversial due to the lack of high-level evidence.Locally advanced TC generally refers to gross extrathyroidal extension (gETE) of the primary tumor that passes the thyroid capsule and infiltrates the surrounding tissues. According to the Cancer Staging Manual of 8th edition American joint Committee on cancer (AJCC), gETE into subcutaneous soft tissues or the larynx, trachea, esophagus, or recurrent laryngeal nerve from a tumor of any size is defined as the T4a category, and the presence of gETE invading the prevertebral fascia, encasing the carotid artery or mediastinal vessels is T4b. than close or positive microscopic margins (R1), while incomplete resection has been associated with increased mortality.,13 In addition, lower local control was observed in recurrent tumors versus initial tumors, and the contribution of postoperative adjuvant radiation therapy was limited. In conclusion, attaining an R0/R1 resection by comprehensive operative clearance is critical and is associated with superior outcomes in T4 disease. In this case, demolitive surgery with reconstruction of the esophagus not only considerably aggravated the notable surgical trauma and postoperative complications but also significantly decreased the patient's quality of life. Above all, the patient's acceptance to undergo such procedures seemed vacillating.Research shows that free margins (R0) have better local control Historically, only a few similar cases were found on review of the literature. Besic et al,15 reported that preoperative ChT decreased the tumor size by >50% in 45% of patients (13/29) with FTC or HCTC and in 44% (7/16) of patients with locally advanced PTC. A case published in 1998 reported that preoperative 131I treatment is beneficial for inoperable TC. In addition, one locally unresectable patient underwent a total thyroidectomy after neoadjuvant EBRT. Nonetheless, a large number of studies have focused on targeted therapies, especially TKIs. For instance, surgical clearance was achieved in patients with locally advanced DTC after 14 months of treatment with sorafenib and lenvatinib, after 13 months of sorafenib monotherapy, or after 22 weeks of lenvatinib monotherapy. Lenvatinib was used to achieve better local control of PDTC during the waiting period for surgery because of patient comorbidities. A similar situation was observed in medullary thyroid cancer (MTC) with sunitinib or even in anaplastic thyroid cancer (ATC) with dabrafenib plus trametinib. Currently, vandetanib and cabozantinib are approved by FDA for patients with progressive, metastatic, or unresectable MTC, while sorafenib and lenvatinib are approved for patients with RAI-refractory DTC. All these findings reveal that neoadjuvant therapy could potentially reduce the extent of invasion and risks of residual disease after surgical resection. In certain cases, it might be a valuable option when treatment is limited.In this particular case, which methods can we try in order to reduce the tumor volume to improve the resection margins and narrow the scope of surgery on the basis of R0/R1 resection? Neoadjuvant therapy for thyroid cancer still has not had an established role and is not mentioned in the 2015 American Thyroid Association Management Guidelines. is a typical representative anti-angiogenesis agent with antineoplastic functions. It could induce apoptosis and suppress tumor proliferation across a variety of advanced solid malignancies with proven efficacy and safety for solid tumors,–31 even TC.,33 Of note, previous studies from our hospital also found that apatinib had better efficacy, a more manageable safety profile and faster therapeutic responses.,34VEGF is a crucial and positive regulator of the physiological angiogenesis processes, which is essential for tumor growth, invasion, and metastasis. VEGFR-2, mainly expressed on endothelial cells, is the key signaling receptor of the pathway mediating the mitogenic, angiogenic, and permeability-enhancing effects of VEGF. Thus, therapies targeting VEGF or VEGFR mainly focus on anti-angiogenesis. Apatinib, also known as Aitan (brand name in China) and developed independently by Shanghai Hengrui Pharmaceutical Co., Ltd. , showed a significant effect and fewer complications in a very short time, consistent with the findings of previous research. Just after treatment for 6 weeks and drug withdrawal for 10 days, R0 resection was accomplished. Tg detection and CT scans were performed to reassess the disease. Fortunately, there were no postoperative complications, such as wound dehiscence or fistula formation. At present, the use of TKIs is limited to thyroid cancer cases that still progress after surgery, radioiodine, or local ablation therapies. There are no data to certify the use of antiangiogenic TKIs as neoadjuvant therapy in DTC. Thus, there are still many issues, including drug categories, dosage, time of adding medicine, withdrawal time before surgery, methods of disease reassessment, and combination with conventional therapies, that are worthy of further study.In this case, the treatment of apatinib To our knowledge, this is the first report that preoperative monotherapy with the VEGFR-2 TKI apatinib was effective, rapid, safe and economical for unresectable locally advanced DTC. The emergence of this neoadjuvant concept has changed the landscape of the management of locally advanced TC. We believe that this study provides a new idea for the treatment of inoperable DTC cases to achieve extensive resection of extrathyroidal tissues and simultaneously improve quality of life. However, more cohort studies, randomized controlled trials, and even related mechanistic research are needed.Resources: Xianzhao Deng, Jie Kang, Bo Wu, Bomin Guo.Writing – original draft: Yingchao Zhang.Writing – review & editing: Zheng Ding, Youben Fan.


We report here a case of an unmarried teenage girl with a 19-kg giant ovarian cystic tumors of size 37cm×31cm×22cm, which was presumably benign on imaging and with negative tumor markers; treated by complete laparoscopic ovarian cystectomy following decompression and the patient was discharged the next day. It is common to encounter cystic abdominopelvic tumors in clinical practice, but it is very rare to find a giant ovarian cyst in today’s world . With th2. On physical examination, she was afebrile with a pulse rate of 84 beats/min, blood pressure of 120/72 mm Hg, and respiratory rate of 24 breaths/min. There was no jaundice, edema, or lymphadenopathy. Secondary sexual characteristics were evident. Abdominal examination revealed a grossly distended abdomen of size bigger than a 36-week gravid uterus that appeared more like ascites with demonstrable fluid thrill as no definite lump could be palpated through the tense abdomen whose girth was 112cm. On percussion, a dull note was appreciated, and no tenderness was noted. Bowel sounds were normal. The external genital examination was normal.A case of an unmarried 19-year-old teenage (nulliparous) girl with giant ovarian serous cystadenoma weighing 19 kg is reported here who presented to Gynecology OPD with gross distension of the abdomen for the past one year. Increasing abdominal girth was accompanied by vague abdominal pain and respiratory discomfort which worsened on lying down. There was accompanying insomnia, anorexia, and weight loss. There was no history of vomiting or other gastrointestinal symptoms, urinary symptoms, colicky pain, or fainting attacks. She had no previous history of illnesses, allergies, or surgery. She denied the use of any medications. There was no family history of malignancies. She had no previous investigations and was from a poor family. Her menarche commenced at the age of 11 years with subsequent regular cycles. Her body weight was 49.5 kg, her height was 151 cm, and her BMI was 21.71 kg/mPregnancy could have been one possibility in a young girl with a distended abdomen which was ruled out as there was no history of amenorrhea. Organomegaly was excluded from clinical examination. Uterine mass was not a possibility as there was no history of menstrual irregularities. So ovarian cyst was the most likely diagnosis.A plain radiograph of the chest was within normal limits. Transabdominal ultrasonography revealed a large abdominopelvic cystic mass extending from the pubis to the epigastrium with a mass effect on abdominal organs and with no evidence of solid components or septations. The uterus was normal and endometrial thickness was 10 mm. Serum CA-125 was 5.4 IU/mL, Serum CEA was 1.5 IU/mL, Serum CA 19-9 was 10.67 IU/mL, Serum Alpha-fetoprotein was 1.3 IU/mL and total HCG was 2.0 IU/mL; all tumor markers were within normal range except serum LDH, which was 685 U/L. Abdominopelvic computerized tomography (CT) findings were consistent with a large well-defined homogeneously cystic lesion 37cm×31cm×22cm  in the abdomen arising from left ovary while right ovary, adnexa, and uterus were unremarkable. Rest all other organs were unremarkable except that left hydroureteronephrosis was noted most probably due to the mass effect of the tumor. As no solid component was noted in the lesion, no abdominopelvic metastases or lymphadenopathy was noted in either ultrasound or CT scan, the possibility of the tumor being malignant was negligible. Considering the cyst to be benign in this unmarried girl, we decided to manage it with laparoscopic cystectomy. Figure After adequate counselling and informed consent, the patient was taken up for laparoscopic cystectomy. Open-entry laparoscopy was performed with an approximately 1 cm incision on the upper border of umbilicus, in which a 10 mm trocar was placed directly into the cyst and a suction catheter was attached to the trocar cannula following which around 16 litres of clear watery cyst fluid was drained. After this the umbilical cannula was repositioned and a stay suture was taken on the cyst wall margins. The telescope was placed via umbilical 10 mm port. A pneumoperitoneum pressure of 12-14 mmHg was achieved and was maintained throughout the procedure. Three ancillary 5-mm trocars were then placed under vision, two on the left side and one on the right side of the umbilical port. The surgery was performed under general anesthesia with the patient in the lithotomy position. Throughout the procedure, the surgical table was put in the Trendelenburg position, but the angle was modified in different phases of the surgery as per the anesthesiologist’s needs. Huge cyst after decompression was still quite big and was seen arising from the left ovary. Right ovary, right fallopian tube and uterus were found to be absolutely unremarkable. Through the laparoscope, the abdominal cavity could only be partially visualized because the giant cyst was in the way and the cyst’s origin was traced with difficulty and it was found to be arising from the left ovary. Following this, cystectomy was performed using bipolar forceps and scissors while conserving the ovarian tissue on the left side. The huge cyst wall was extracted completely through the 10-mm umbilical port, and it weighed about 1,150 g. A 5-mm telescope was used which was introduced through one of the side ports. A 10-mm grasper was then introduced through the 10-mm umbilical port and cyst wall was grasped under vision and brought outside the umbilical skin incision for extraction. The final volume of the drained cyst fluid content was 15,950 mL. Afterward, pneumoperitoneum was induced again, and systematic visual inspection of the abdominal cavity was done. The liver, gallbladder, spleen, and diaphragm appeared normal, and no macroscopic signs of malignancy were noted. No blood loss or other intraoperative complications occurred. The total operative time was 150 min. Patient tolerated the procedure well and had an uneventful recovery. Figure The patient started to ambulate on the same day of surgery, passed urine after self-retaining catheter was removed in 12hrs and was discharged the next day. No malignant cells were reported on cytology examination of the cyst fluid and the cyst wall was reported as serous cystadenoma on histopathology Figure .Ovarian tumors involve a variety of histological tissues, of which epithelial tissues are the most common. Eighty percent of all epithelial tumors are benign. Serous cystadenomas are the most common cystic ovarian tumors. In the above case, probably due to non-troubling symptoms, the patient did not seek medical attention early which led the tumor to enlarge and reach a giant size. Socioeconomic factors also contributed to her late medical consultation. The absence of solid nodules, ascites, and increased vascularity on imaging and negative tumor markers pointed toward a diagnosis of a benign tumor, which helped in deciding the choice of surgery for this patient. Laparoscopic cystectomy with no spillage and preservation of ovarian tissue is the management of choice in a benign ovarian cyst where fertility is desired, and this is what was done in the above case. There is no clear consensus or proper guidelines regarding the upper limit of the size of the tumor that can be operated via minimally invasive surgery as there is a risk of capsular rupture, peritoneal spillage, and seeding in case the tumor turns out to be malignant or borderline. However, the advantages of laparoscopic surgery such as lesser postoperative pain, shorter hospital stay, better cosmetic outcomes, and early return to activities in addition to the advancing techniques of specimen retrieval in minimally invasive surgery to tackle peritoneal seeding have led to its increased preference over open surgery in benign cases.To date, few such cases have been reported in the literature. Dolan et al. reported a giant ovarian cyst of 40.5cm×30cm×22.5cm which was decompressed by minilaparotomy followed by laparoscopic oophorectomy . Sanna eTo our knowledge, this is the first case reported of complete laparoscopic management of a giant ovarian tumor more than 35 cm in the largest dimension where minilaparotomy was not done to remove the tumor.To conclude, complete laparoscopic surgery with decompression can be done for giant ovarian cyst removal provided the tumor seems benign on comprehensive workup. However, care should be taken to avoid spillage during decompression. Considering the advantages of minimally invasive surgery, the size of the tumor should not be a limiting factor in attempting minimally invasive surgery in seemingly benign ovarian cystic tumors. Various different ways and techniques for specimen retrieval have been described as not necessitating minilaparotomy. However, one should not also be overzealous in attempting laparoscopic surgery in a case where malignancy is not completely ruled out.


Pancreatic neuroendocrine carcinoma (PanNEC) is a rare disease entity with rapid progression and poor prognosis. Here, we report a PanNEC case with unique morphological features mimicking intraductal papillary mucinous carcinoma.A 69-year-old Japanese man was referred to our hospital for further evaluation of weight loss and deterioration of diabetes mellitus. Contrast-enhanced computed tomography showed a solid and cystic mass with hypo-enhancement at the tail of the pancreas. The main pancreatic duct (MPD) was diffusely dilated without obstruction, accompanied by marked parenchymal atrophy. Multiple peritoneal and omental nodules were observed, suggesting tumor dissemination. Endoscopic retrograde cholangiopancreatography revealed that the mass correlated with the dilated MPD. During pancreatography, a large amount of mucus was extruded from the pancreatic orifice of the ampulla. Based on these imaging findings, intraductal papillary mucinous carcinoma was suspected. Per-oral pancreatoscopy (POPS)-guided tumor biopsies were conducted for the lesion's solid components. Histopathological examination of the biopsied material confirmed small-cell-type PanNEC with a Ki-67 labeling index of 90%. Due to his condition's rapid decline, the patient was given the best supportive care and died 28 days after diagnosis.Although rare, PanNEC, which correlates with the MPD and is accompanied by marked dilation of the MPD, does exist as one phenotype. In such cases, POPS-guided biopsy could be a useful diagnostic modality. Pancreatic neuroendocrine neoplasm (PanNEN) is a rare type of pancreatic neoplasm, accounting for 1%−2% of pancreatic tumors . TreatmeDue to its rarity and non-specific imaging findings, PanNEC is sometimes difficult to consider as a differential diagnosis for various pancreatic neoplasms; therefore, histopathological examination is mandatory for confirmatory diagnosis. Endoscopic ultrasonography-guided tissue acquisition (EUS-TA) has been adopted as a first-line procedure for the definitive histological diagnosis of pancreatic lesions , 8. ReceHere, we report a PanNEC case with cystic degeneration masquerading as intraductal papillary mucinous carcinoma (IPMC), which was successfully diagnosed by direct tumor biopsy under POPS.2 and hypointensity on the apparent diffusion coefficient map, indicating diffusion restriction , C-reactive protein , and decreased total protein and albumin levels. Tumor markers showed a mild elevation in carcinoembryonic antigen , carbohydrate 19–9 , and a marked elevation in neuron-specific enolase . The level of duke pancreatic monoclonal antigen type 2 (DUPAN-2) was within the normal limits. Abdominal ultrasonography showed a well-defined, irregular-shaped, heterogeneous mass at the pancreatic tail . The MPDtriction . The sigtriction , suggesttriction . AdditioThe features of the pancreatic tumor with cystic components that were communicated with the MPD favored the possible diagnosis of IPMC. Since the MPD was diffusely dilated, POPS-guided direct tumor biopsy under endoscopic retrograde cholangiopancreatography (ERCP) guidance was planned to confirm the histological diagnosis. The ampullary orifice was dilated to the so-called fish-mouth appearance . After EDue to the patient's rapid deterioration, chemotherapy could not be initiated. Although the best supportive care was given to the patient, he died 28 days after the diagnosis.We reported a case of PanNEC with interesting imaging findings mimicking IPMC. Based on their morphological characteristics, pancreatic tumors are roughly classified into solid and cystic neoplasms. PanNECs are typically solid neoplasms, whereas IPMCs are some of the most common pancreatic cystic tumors; thus, imaging findings of the two are quite different. However, PanNECs can cause cystic change, which sometimes makes it difficult to distinguish from other pancreatic cystic tumors, as in this case. The frequency of cystic change in PanNEN is about 11%−17% . It has One question that arises from this case is whether the large amount of highly viscous fluid in the MPD was produced from the PanNEC tumor cells. Conceptually, since PanNENs do not produce mucin, two major hypotheses are considered: 1) coexistence of other mucus-producing tumors represented by intraductal papillary mucinous neoplasm (IPMN) or 2) trans-differentiation to neuroendocrine cells from other mucin-producing tumor cells. As for the former hypothesis, although whether the coexistence of IPMN and PanNEN is a real association or a coincidence remains unclear, the association between the two neoplasms has been increasingly reported , 16. To Regarding radiological findings, PanNECs are characterized by hypo-enhancing masses that show heterogeneous enhancement, sometimes accompanied by cystic changes, calcification, and necrosis . FollowiCurrently, EUS is considered a well-established diagnostic modality for differentiating pancreatic tumors because of its high-resolution images obtained in real time . NotablyVisually directed biopsy with POPS is another modality for the histological diagnosis of pancreatic tumors originating from the MPD and tumors communicating with the MPD. A recent study that included 78 cases reported that the sensitivity, specificity, and diagnostic accuracy values for visually directed biopsies under POPS guidance were 91%, 95%, and 94%, respectively . Althougvs. 7 months without surgery)  . In recesurgery) , 32. Regsurgery) , but in surgery) . AccordiIn conclusion, we have reported a case of PanNEC mimicking IPMC. Although rare, it should be noted that PanNEC, which correlates with the MPD and is accompanied by marked dilation of the MPD by highly viscous fluid production, does exist as one phenotype. Therefore, histological diagnosis is mandatory for the diagnosis of PanNEC with atypical imaging findings, and this case highlights that POPS-guided biopsy can be a useful diagnostic modality. However, the accumulation of additional data from more cases is necessary to further elucidate this type of PanNEC.The original contributions presented in the study are included in the article/supplementary material, further inquiries can be directed to the corresponding author.The studies involving human participants were reviewed and approved by the Ethics Committee of the Kindai University Hospital. The patients/participants provided their written informed consent to participate in this study.HT and KM wrote this manuscript, performed the endoscopic procedures, and cared for the patient. TH and KM analyzed and interpreted the imaging findings. MKi performed pathological examinations. YO, KK, KY, and MT reviewed the literature. TW and MKu critically revised the manuscript for intellectual content. All authors have read and approved the final manuscript.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.


Osteochondromas are bone lesions composed of medullary and cartilaginous bone covered by a cap of hyaline cartilage. The presence of medullary and cortical bone with the continuity of the tumor is pathognomonic for osteochondroma and aid in establishing the diagnosis.We report a case of a two-year-old girl who presented to our clinic following her mother noticing a palpable, growing, and painful mass on her left scapula. There was no limitation in the range of motion. A clear-cut mass was seen on the dorsal aspect and palpated measuring around 2.5x3 cm. Surgical excision of the mass followed by histologic examination confirmed osteochondroma. Upon follow-up, the patient had no pain and had a full range of left shoulder motion without discomfort or pain.In conclusion, scapular exostoses are very rare and more so when they present dorsally. Symptomatic lesions can be managed effectively with surgical excision of exostosis. Osteochondromas are bone lesions composed of medullary and cartilaginous bone covered by a cap of hyaline cartilage. The presence of medullary and cortical bone with the continuity of the tumor is pathognomonic for osteochondroma and aid in establishing the diagnosis -4. They We report a case of a two-year-old girl who presented to our clinic following her mother noticing a palpable growing and painful mass on her left scapula for 18 months. No limitation in the shoulder and scapulothoracic joint's range of motion was appreciated. No similar history among other family members was present. A well-defined round mass was seen on the dorsal aspect and palpated measuring 3 x 2.5 cm (Figure The mass was severely painful, especially in the supine position. A CT scan confirmed the diagnosis of osteochondroma Figure .A nuclear bone scan was done, which concluded the presence of focal uptake in the left scapula corresponding to moderate osteoblastic activity with regular contours and mild sclerotic changes, which were likely related to exostosis, with no other lesions in the body noted on the scan. X-rays showed a bony growth on the dorsal aspect of the scapula Figure .The decision to go for surgical removal with a safety margin was made. The patient was placed in a prone position under general anesthesia. An incision was done right above the mass, separating the muscle directly from the mass, and excising the mass from the base so that no residual part of the mass is left behind. The stalk of the exostosis was excised at the base with an osteotome from the dorsal surface of the scapula Figure . The speThe patient improved immediately in terms of pain and was followed up in the clinic regularly. Within almost a year, the patient had no pain, and had a full range of left shoulder motion without discomfort or pain. Follow-up X-rays showed no evidence of recurrence Figure . The patAs noted previously, osteochondromas of the scapula account for 4% of all bone tumors occurring in the scapula ,43. MostMoreover, some cases reported worsening pain Table  42,48,5,51,59, aGenerally, osteochondromas are managed after skeletal maturity to avoid injuring the growth plate during surgery ,71, giveOsteochondromas of the scapula, although benign, are at risk of being left unnoticed until malignant transformation occurs, like other central osteochondromas; therefore, we routinely advocate the removal of scapular osteochondromas at presentation. Excision after diagnosis can be done in a meticulously planned manner to avoid iatrogenic injury to the growth plate and ensure complete excision at the base of the stalk. The patient’s family is very grateful for the full recovery of their daughter and is satisfied with the results.


Tumor could directly invade or is adherent to other organs, but superior mesentery vein (SMV) and duodenum invasion are very rare. A 62-year-old woman was diagnosed with abdominal pain for several months. Multiple erythematous brownish skin patches and palpable mass were found at epigastric area. Computed tomography imaging showed focal wall thickening at the transverse colon that invaded to the rectus muscle and anterior abdominal wall. On exploration, we identified tumor invaded or was adherent to the duodenum and superior mesenteric vein and performed en-bloc resection. After surgery, the patient received chemotherapy and was followed up without any recurrence for 16 months. Adhesion and invasion of tumor to surrounding organs can be unexpectedly found during surgery. In our case, we found duodenum and SMV invasion and achieved R0 resection by SMV and duodenum resection, which could improve the patient’s prognosis. Locally advanced colon cancer (LACC) is clinically defined as primary colon cancer with direct invasion to the adjacent structures or extensive regional lymph node involvement. Incomplete excision of the tumor and residual tumor in the neighboring organs are risk factors for poor prognosis and recurrence in LACC. En-bloc radical colectomy initially proposed by Bacon still remains controversial and is a challenging surgical matter . SeveralA 62-year-old woman was referred to the emergency room with abdominal pain. She had hypertension and had undergone right hemicolectomy due to colon cancer 22 years ago. A physical examination revealed multiple erythematous brownish skin patches and palpable mass at epigastric area . All rouShe underwent computed tomography (CT), which showed focal wall thickening at the transverse colon with direct invasion to the rectus muscle and subcutaneous fat layer of the anterior abdominal wall . The surWe made an incision avoiding the tumor. The patient’s abdominal cavity showed overall adhesion due to previous surgery. We found tumor invading or being adherent to the duodenum and superior mesenteric vein . We thouThe pathology results revealed a T4bN1b moderately differentiated adenocarcinoma, negative resection margins, and positive lympho-vascular and perineural invasion at transverse colon and a T1N0 moderately differentiated adenocarcinoma, negative resection margins, positive lymphatic invasion at sigmoid colon. The patient’s postoperative course was uneventful and she was discharged on the 23rd post-operative day. After surgery, she received FOLFOX chemotherapy and was being followed up without any recurrence for 16 months.At the time of diagnosis, 10–20% of patients have LACC, which means that the tumor has penetrated the bowel wall and infiltrated neighboring organs. Historically, patient with an LACC was expected to have a poor prognosis. Recent studies, however, report that their survival can be comparable to that of patients with less advanced tumors if effective treatment is administered , 6. In aAmong the multi-visceral resections for LACC, jejunal and ileal resections are performed most often and duodenum and SMV resection are extremely rarely performed. The peritoneum (35%), abdominal wall (25%), small bowel (other than the duodenum) (16%), omentum (16%), urinary bladder (14%), and ovaries (12%) were the most frequently invaded adjacent structures; invasion of the duodenum (3%) uterus (4%) stomach (4%) retroperitoneum (4%) and Gerota’s fascia (2%) was uncommon . InvasioThanks to the development of CT technology, lymph node and distant organ metastases can be evaluated with high accuracy and the diagnosis of LACC is possible pre-operatively . CT can Infiltration in surrounding organs can be unexpectedly found during surgery, however, proper resection may not be performed due to the patient’s advanced age and the surgeon’s lack of experience. Without R0 resection, 5-year survival rate ranges from 0 to 23% , 12. HowIn conclusion, adhesion and invasion of adjacent organs can be unexpectedly found during locally advanced colorectal cancer surgery. In our case, we found duodenal and SMV invasion during surgery and achieved R0 resection by SMV and duodenal resection, which could improve the patient’s prognosis.


Infantile hemangiomas are characterized as benign tumors of vascular tissue that arise from rapid endothelial cell proliferation followed by gradual involution, affecting 4% to 5% in infants and 2.6% to 9.9% in older children. Most of them resolve by the age of three years, negating the need for surgical intervention. However, intervention should be considered especially in cases with a high risk of recurrence. A female patient, aged 10 years, was referred to plastic surgery by her dermatologist due to the presence of a vascular mass in her face located at the junction between the nose and right cheek that had been present since infancy. The patient was diagnosed with infantile hemangioma based on MRI imaging of the face showing a benign vascular lesion measuring 9 x 12 mm. After the failure of multiple sclerotherapy sessions and informed discussion with the respective family, the patient underwent open rhinoplasty for surgical excision with no facial scarring other than the transcellular scar. This study presents a rare case of utilizing the open rhinoplasty technique in a relapsing facial hemangioma of a 10-year-old child. Results show a positive aesthetic outcome by minimizing facial scars. Considering the limited reported use of this technique, more clinical studies, especially comparing long-term effects across different age populations, are recommended to validate the efficiency and effectiveness of this technique. Infantile hemangioma (IH) is the most common pediatric vascular tumor. It is estimated to affect 4% of infants though the reported incidence ranges from 2-10% [IH usually occurs during the first few weeks of life and follows a typical natural path of proliferation and involution. Their clinical appearance depends on their depth and distribution. Its pathogenesis is not well-known but is thought to represent an abnormal response of pluripotent stem cells to stimuli such as hypoxia and the renin-angiotensin system.Classification comprises superficial, mixed, and deep IH as well as IH with minimal or arrested growth.Large facial or lumbar IH permits investigation for the PHACE  and LUMBAR  syndromes, respectively. The complications of IH include ulceration, obstruction or functional impairment, hypothyroidism, and cosmetic sequelae.In this case report, we present a child with facial hemangioma treated by open rhinoplasty.Our patient was a 10-year-old Saudi female who presented to the senior author (MAM) and was diagnosed with congenital nasal hemangioma. She is the third child of healthy parents. The pregnancy was uncomplicated, and the family history was negative for any congenital anomalies. Prior to her presentation, the patient was treated previously by oral propranolol by her dermatologist. On clinical examination, the patient had a vascular mass on the face located at the junction between the nose and right cheek Figures -1C. RadiThe typical presentation of infantile hemangioma is a solid vascularized tumor with cutaneous lesions that may present as red patches on any part of the body and usually appear after two weeks of age . The diaUsing the open rhinoplasty approach for excision in the pediatric group is considered challenging. The key to ideal reconstruction is the preservation of skin and as much healthy soft tissues as possible, as well as avoiding steroid use, topically or systemically along with cryotherapy preoperatively, as it disrupts the integrity of the skin . In addiHemangiomas are very common in the pediatric population with several management options. This study presents a rare case of utilizing the open rhinoplasty technique in removing and reconstructing the facial hemangioma of a 10-year-old patient. This approach suggests a positive aesthetic result in minimizing facial scarring after surgery. Considering the limited report of this approach, further clinical studies especially comparing the long-term effects across different age populations, are strongly recommended in reporting this technique, to describe and validate its efficiency and effectiveness.


Lumbar hernia is an uncommon condition that can either be congenital or acquired. Acquired lumbar hernia is further divided into primary, with no identifiable cause, and secondary, occurring due to previous trauma, infection, or surgery. Here, we present the case of inferior lumbar hernia in a 65-year-old Asian male who was a chronic alcoholic and smoker. He presented with a complaint of a longstanding swelling in the right lumbar region for five years and no other associated symptoms. The swelling was reducible, an expansile cough impulse was felt on palpation, and bowel sounds were heard on auscultation. A contrast-enhanced computed tomography scan revealed a 6.7 cm defect in the lateral abdominal wall in the right lumbar region with bowel loops, cecum, ascending colon, mesentery, and mesenteric artery seen herniating through the defect. There was a history of an iliopsoas abscess at the same site five years ago, which was treated with incision and drainage. The patient was advised for an open mesh repair but could not be operated upon due to coexisting aortic stenosis and regurgitation. Our impression, from this report, is that a chronic iliopsoas abscess tracking to the inferior lumbar region and the incision and drainage thereof, leading to a weakness in the abdominal wall, may be considered to be a cause of inferior lumbar hernia, with chronic smoking on part of the patient being a significant contributing factor for the abdominal muscle weakness. Therefore prompt and meticulous treatment of an iliopsoas abscess must be done to prevent this complication. Lumbar hernia is a rare entity with approximately only 300 case reports published worldwide . The lumLumbar hernias can be congenital 10-20%), arising from congenital muscle weakness, or acquired (80-90%), which can be spontaneous, also known as primary, or because of any preceding events such as trauma, infection, or postoperative, also known as secondary acquired hernia 0%, arisi. Lumbar We report a case of unilateral right-sided inferior lumbar hernia secondary to an iliopsoas abscess encountered in a 65- year-old male who was a chronic alcoholic and smoker. The patient could not be operated upon because of the coexisting moderate aortic stenosis (AS) and aortic regurgitation (AR).This case report was previously presented as an abstract at Revolutionizing Global Surgery, an annual Global Surg Network international conference on November 14, 2021.A 65-year-old Asian male presented to the surgery ward with a chief complaint of swelling in the right lumbar region for five years. The swelling was sudden in onset and had gradually increased in size over the years. The swelling was self-reducible, reducing on lying down in the left lateral position and reappearing on lying down supine or standing. There was no history of any previous trauma. There was no history of pain or fever associated with the swelling, which ruled out the possibility of strangulation. The patient was a chronic alcoholic and smoker for the past 30 years with two to three packs smoked per day.Mycobacterium tuberculosis was negative on investigations. There was a delay on part of the patient to seek medical care. The abscess was later treated with incision and drainage.The patient reported a history of an iliopsoas abscess at the same site five years ago, the etiology of which was not found. Clinical examination revealed a swelling measuring 30 cm × 15 cm in the right lumbar region Figures , 1B. A sUltrasonography of the abdomen revealed a 5.5 cm × 1.5 cm defect in the right lateral abdominal wall, with mesentery and bowel loops seen herniating through it. A coincidental finding of a left-sided renal cyst 4.9 × 5.7 cm was also seen.Contrast-enhanced computed tomography (CECT) scan of the abdomen revealed a few small, hypodense, non-enhancing cystic lesions in segments IV and V of the liver, the largest measuring about 15 × 18 mm in size, suggestive of hepatic cysts. Few cortical cysts were seen in bilateral kidneys, the largest measuring 5.1 × 3.8 cm in the upper pole of the left kidney. As seen in Figures Echocardiography showed grade one diastolic dysfunction with global hypokinesia, predominantly of the mid and distal septum, inferobasal left ventricular wall, and left ventricular apex. Mild septal hypertrophy and concentric left ventricular hypertrophy were present. The aortic root measured 2.4 cm. The aortic valve was thickened and restricted in motion. The maximum pressure gradient across the valve was 52 mmHg and showed trivial AG and moderate AS. The left ventricular ejection fraction was 55%.Depending on the clinical and radiological findings, a diagnosis of right-sided lumbar hernia was made. Based on the findings, the patient was advised for an open mesh repair. He was referred to a cardiologist for the coexisting AG and AS and was recommended aortic valve replacement before proceeding with the open mesh hernia repair; however, the patient elected to avoid the surgery due to financial constraints. The patient is being followed up fortnightly, and there are no fresh complaints of skin changes or pain at the hernia site.Lumbar hernia is an uncommonly encountered condition that can be divided into two categories, namely, superior and inferior lumbar hernia. It usually presents as a swelling in the lumbar region. While it is mainly asymptomatic, it may present with pain, backache, or strangulation, presenting as an acute abdominal emergency ,5. Our cLumbar hernias can easily be diagnosed based on clinical and radiological findings, with a CT scan being the gold standard . While mThe preferred treatment of a lumbar hernia is an open prosthetic mesh repair, but laparoscopic mesh repair is being increasingly considered . The onlLumbar hernias are extremely rare and present as a swelling or mass in the lumbar region. They can be diagnosed based on clinical and radiological findings, with a CT scan being the gold standard. A lumbar hernia occurs most commonly due to secondary causes such as trauma, infection, or surgery. Through this report, we conclude that a chronic iliopsoas abscess tracking to the inferior lumbar region and the incision and drainage thereof, leading to a weakness in the abdominal wall, may be considered as a cause of the inferior lumbar hernia, with chronic smoking on part of the patient being a significant contributing factor for the abdominal muscle weakness. Treatment varies depending on the patient variables and needs to be individualized. Mesh repair of the hernia is the most commonly preferred method which can be done either laparoscopically or by open repair. Primary repair of the defect is usually not preferred in a large hernia because of the high risk of recurrence. We report this case because a lumbar hernia secondary to an iliopsoas abscess has not yet been documented in the literature and more studies investigating this complication are required.


Liposclerosing myxofibrous tumor (LSMFT) is a rare benign fibro-osseous tumor that most frequently occurs in the proximal femur. The reported literature shows that the proximal femur, ilium, tibia, humerus, rib, and skull have occurred, but so far, the female distal femur has not been characterized in detail. This, we think, is the first single comprehensive case report of the female distal femur. To prevent misdiagnosis and overtreatment of this illness, it is critical for us to continue strengthening our knowledge of it and to add it to the differential diagnosis of the space-occupying lesion of the female distal femur.Two months ago, a 55-year-old female patient was found to have a space-occupying lesion of the left distal femur and the pain symptom was aggravated. She underwent thorough curettage and bone grafting without additional treatment to relieve the current symptoms and determine the nature of the lesion in our hospital. The intraoperative specimens were submitted to the pathology laboratory for analysis, and the result was reported as LSMFT. And six months after the operation, the patient returned to our hospital for another x-ray examination and we found that she had recovered well without any signs of recurrence. The patient self-reported that she had now resumed her daily life without any uncomfortable symptoms.The incidence of LSMFT itself is relatively low, and the occurrence of the distal femur is even rarer. However, it is recommended to add LSMFT into the differential diagnosis of the occupying lesions of the distal femur. Once the diagnosis is made, thorough curettage and bone grafting without additional special treatment can achieve better postoperative outcomes. The patient gave her agreement after learning that information about the case will be submitted for publication. Liposclerosing myxofibrous tumor (LSMFT) was first described by Ragsdale and Sweet in 1986 . The conIn this rare case report, we document in detail a female patient who was found to have a space-occupying lesion in the distal femur and was diagnosed with LSMFT. She was treated only by complete curettage, and bone grafting and recovered satisfactorily. We think this may be the first case of LSMFT in the female distal femur that has been described in detail. We document the clinical, radiological, and pathological features as well as a retrospective discussion and summary of the case report.A 55-year-old female patient went to a local hospital for a magnetic resonance imaging (MRI) examination of her left knee 2 months ago because she felt swelling and dull pain in her left knee after daily walking. The result showed a space-occupying lesion in the left distal femur. The local doctor suggested conservative treatment and regular assessment. Conservative therapy lasted for one month, during which the symptoms did not improve, but the distension and pain of the left distal femur became more apparent, so she went to the orthopedic clinic in our hospital. She was found to have pain on the lateral side of her left distal femur by pressing, without touching the mass during the physical examination. The skin temperature of the left distal femur was normal and the skin could move freely. Anteroposterior and lateral radiographs of the left knee showed a patchy high-density shadow of the left distal femur without fracture or bone destruction . MRI of α mutation is also reported in two patients who are diagnosed with LSMFT, which is the similar mutation found in cases of fibrous dysplasia. Therefore, the authors of this paper believe that LSMFT is closely related to fibrous dysplasia (Liposclerosing myxofibrous tumor (LSMFT) was first described in 1986 and is most likely to occur in the proximal femur, accounting for 80%–90% of the cases , 7. Someysplasia . But somysplasia , 14. Sevysplasia , 4, 15. ysplasia , 4, 5, 7ysplasia . Althougysplasia , 16–18. ysplasia , 19. Theysplasia , 9, 15. ysplasia . CampbelLSMFT has exhibited a considerable preference for the proximal femur. At the same time, ilium, humerus, ribs, and skull have also been reported. On the other hand, we have not found any literature detailing the occurrence of LSMFT in the female distal femur. This lesion often has a sclerotic border and is often a well-defined geographic lytic lesion. However, anteroposterior and lateral radiographs of the left knee showed a patchy high-density shadow of the left distal femur without fracture or bone destruction in this case. The diagnosis of this rare disease depends on a comprehensive analysis of clinical, radiological, and histopathological findings. The tumor may be readily mistaken for other lesions with possibly overlapping characteristics because of the diversity of its tissue structure. This rare case report is used to expand our awareness of LSMFT in the distal femur and to remind us to add LSMFT to the differential diagnosis before making a diagnosis of the space-occupying lesions of the female distal femur, with the final pathological diagnosis as the gold standard. Once the diagnosis is confirmed, thorough curettage and bone grafting are carried out without additional treatments. x-ray or MRI examinations are regularly ordered after the operation, and the patient's recovery status is closely followed. Generally, the postoperative recovery is excellent without affecting daily life.


Penile cancer is generally rare, and Squamous cell cancer of the penis is the most common histological type. Sarcoma of the penis has a low incidence, but they tend to grow faster than other penile cancers. One of the rarest types of penile sarcomas is Extra-Skeletal Ewing's Sarcoma (EES). The management of such cases can be challenging, and treatment guidelines do not exist for these rare cases. We present a rare case of EES that has developed in the penis of a young patient in the United Kingdom. Ewing's sarcoma (ES) is a rare tumor; however, it is considered the second most common bone tumor of malignant nature in children . It fallIf ES primarily arises from soft tissue, it is called Extra-skeletal Ewing's sarcoma (EES). It represents 10-20% of ES, commonly arising from extremities, chest wall, paravertebral space, and retroperitoneum. Rare locations were also reported, such as the heart, kidneys, jejunum, larynx, and vulva . PrimaryAn 18-year-old male patient, who does not have any significant past medical or family history, presented to his local hospital with a progressively enlarging painless lump at the base of the penis on the left side over three months. It has not affected his urinary or erectile function, and he did not report any other systemic symptoms such as weight loss or loss of appetite.Clinical examination revealed a 6 cm firm mass at the penoscrotal junction, which felt attached to the underlying penile structures. Both testicles were spared and of normal size and consistency, and there was no palpable regional lymphadenopathy.Magnetic Resonance Imaging (MRI) of the penis has been arranged. It showed a 5.5 cm enhancing mass at the base of the penis that appeared separate from the underlying corpora cavernosa Figures -3. StagiAn incision biopsy was performed. Immunohistochemistry revealed tumor cells that showed positivity with CD99, focal positivity with CD56, and negativity with CD45, CD20, CD3, S100, myogenin, WT1, MYO-D1, and myeloperoxidase. Scattered cells also express Epithelial Membrane Antigen (EMA). The features of a poorly differentiated round cell sarcoma were consistent with a soft tissue Ewing's sarcoma. This diagnosis has been confirmed by demonstrating a characteristic EWSR1 (22q12.2) rearrangement detected by Fluorescence In Situ Hybridisation (FISH). Given the rarity of the case, the histopathological diagnosis was challenging. The case was sent for an expert pathologist review in a highly specialized orthopedic center, which agreed with the diagnosis of EES. A staging fluorodeoxyglucose (FDG) Positron Emission Tomography-Computed Tomography (PET/CT) scan followed and confirmed FDG avid lesion of the penis in keeping with the known sarcoma. No FDG avid metastatic disease was demonstrated; however, some mildly prominent retroperitoneal lymph nodes were believed to be reactive rather than malignant.The case was reviewed in the Supra-Regional Penile Cancer multi-disciplinary team meeting (MDT), which questioned the involvement of the urethra as it was not clear on the initial MRI scan. The imaging review was suspicious for tumor extension beyond the pubic symphysis. According to Ewing's sarcoma protocol, the patient received neo-adjuvant chemotherapy, which involved 11 cycles of alternating Vincristine/Doxorubicin/Cyclophosphamide and Ifosfamide/Etoposide. He was also offered sperm storage. He had an excellent response to the neo-adjuvant chemotherapy with significant downsizing of the tumor seen on serial imaging Figures -6.The patient then underwent radical total penectomy and perineal urethrostomy, which included an elliptical incision around the base of the penis, mobilization of the penis 4cm proximal to the mass, and routine hemisection of the penis and the urethra 1.5cm proximal to the mass than the creation of a perineal urethrostomy. Leaving an adequate stump for consideration of surgical reconstruction while ensuring adequate excision with clear margins is the challenge of the operation.The specimen was sent for histopathological assessment Figures -10. PostHistopathology confirmed that the morphology and immunophenotype are compatible with the original diagnosis of Ewing's sarcoma, with chemotherapy-induced fibrotic changes of the adjacent stroma. The surgical margins were confirmed to be clear. According to the protocol for follow-up of intermediate and high-grade soft tissue sarcoma, the patient will have three monthly follow-ups for one year, four monthly follow-ups until year 3, 6 monthly follow-ups until year five, then yearly follow-ups for 10 years, and then discharge can be considered. The follow-ups might include regular MRI or CT scans and chest X-rays depending on the patient's symptoms and the judgment of the oncologist or the surgeon.Extraskeletal occurrence of ES is rare; however, it can occur in any location . Upon reThe diagnosis is made with imaging, and a biopsy obtains a tissue diagnosis. MRI scan with prostaglandin injection to artificially induce erection is the imaging modality of choice for penile lesions . MRI feaHistologically, EES is composed of round or oval cells with round hyperchromatic nuclei separated by fibrous septa. These cells are usually rich in glycogen . ImmunohEES is chemo and radiosensitive. Treatment is, therefore, multi-modal, neo-adjuvant chemotherapy and surgical treatment with or without adjuvant radiotherapy . PsycholThe occurrence of primary penile EES is infrequent. Our case exemplifies how a multi-disciplinary approach in a specialized penile cancer center is essential in providing optimum oncological, functional, and psychological care and outcomes in such rare cases. As it is a rare case, no guidelines for management are available. Our case highlights the required investigations for diagnosis and the steps of management. It also emphasizes the role of psychological support, especially for young patients, and the importance of taking future reconstruction into account.