Optic nerve hemangioblastoma is a very rare benign tumor with only 39 reported cases by now. It appears to be hyperintense on T2-weighted images with a significant enhancement on contrast scans, which are similar to glioma and meningioma. Due to the lack of specificity in MRI manifestations, optic nerve hemangioblastoma is often misdiagnosed. To provide new insights into differential diagnosis of optic nerve hemangioblastoma, we report for the first time an optic nerve hemangioblastoma case employing advanced magnetic resonance techniques including diffusion-weighted imaging (DWI), apparent diffusion coefficient (ADC) maps, and magnetic resonance angiography (MRA). In addition, we have collected all reported optic nerve hemangioblastoma cases and reviewed their neuroimaging findings by MRI and angiography. Our results show that solid-type tumor is the dominant form of optic nerve hemangioblastoma and extensive edema is widely observed. These findings are surprisingly contrary to manifestations of cerebellar hemangioblastoma. Besides the structural features, quantitative indexes including ADC and relative cerebral blood volume (rCBV) ratio, which are significantly elevated in cerebellar hemangioblastoma, may also shed a light on the preoperative diagnosis of hemangioblastoma of optic nerve. Finally, we discuss the critical neuroimaging features in the differential diagnosis between optic nerve hemangioblastoma from optic pathway glioma and optic nerve sheath meningioma. Hemangioblastoma is an uncommon benign tumor of central nervous system (CNS), accounting for 2% of all CNS tumors and 7%–12% of all posterior fossa tumors . VHL synOptic nerve hemangioblastoma (ONH) is a very rare subtype with only 39 cases reported. Unlike hemangioblastoma in cerebellum and other locations, most ONH (71%) were found to be associated with VHL syndrome. Visual defects and exophthalmos are the most common symptoms . HoweverHere, we employ advanced MR techniques including diffusion weighted imaging (DWI), apparent diffusion coefficient (ADC) maps and MR angiography (MRA) in an ONH case, expecting to offer more imaging information on the preoperative diagnosis. To our knowledge, it is the first reported ONH case characterized by these advanced MR techniques. Also, we have collected all other published ONH cases, summarized their neuroimaging findings, and compared them with other easily confused diseases of optic nerve such as optic pathway glioma and optic nerve sheath meningioma.A 41-year-old male with a past history of cerebellar hemangioblastoma complained of vision loss in the left eye for 6 years, with reduced vision acuity in the right eye for 3 months. His vision acuity in the left eye started to deteriorate 6 years ago without other discomforts. CT scans at that time showed a mass at cerebellum. He underwent a resection surgery but the left eye still went blind several months later. Postoperative pathological examination confirmed the diagnosis of hemangioblastoma. Two years ago, MRI revealed a newly occurring mass adjacent to the left optic nerve which was asymptomatic. Unfortunately, the follow-up visits showed that it had been progressing gradually without treatment.Supplementary Material). Taken together, these imaging results highly suggested glioma. Hemangioblastoma was also suspected considering his past history. The latest MRI showed another mass in the left cerebellum, which was hyperintense on T2-weighted imaging (T2WI) and hypointense on T1-weighted imaging (T1WI) and DWI, with significant enhancement on contrast scans. These MRI findings and the location of posterior fossa strongly supported the possibility of hemangioblastoma. Combining these imaging clues and his past history, the recurrence of hemangioblastoma at both cerebellum and optic nerve was highly suspected.Three months ago, his right eye had a significant reduction in vision acuity from 20/20 to 20/25 and was referred to our hospital. The visual field test and fundoscopy revealed no abnormality in the right eye. MRI revealed a mass occupying from the left orbit apex to the anterior cranial fossa, with hypointensity on T1-weighted images and hyperintensity on T2-weighted images  showed a complete excision of the mass. The edema has receded postsurgery but not completely. Changes in vision or visual fields have not been observed during the 2-week hospital admission after surgery.The patient then received a left frontotemporal-approach craniotomy to resect the mass. At surgery, a reddish tumor adherent to the left optic nerve was observed. The attached tortuous vessels originated from ophthalmic artery indicated its rich blood supply. The tumor was excised and measured 1.5 × 0.8 × 0.8 cm. Postoperative MRI  and GFAP(+), and the vessels are CD34(+), CD31(+), and F8-R(+). Interspersed CD68(+) monocytes and macrophages were also observed. CgA and Syn were both negative among the whole tissue Figure 2We collected all 39 published ONH cases and reviewed their neuroimaging results.MRI findings of ONH are shown in The typical form of cerebellar hemangioblastoma is a large cyst with mural nodules, accounting for 54.8%, while the solid form composes about 28.8%  is another superior method derived from MRI. Hakyemez et al.  and She In the early time when MRI has not been widely used, angiogram was a useful tool for the diagnosis of brain lesions and identifying feeding arteries and draining veins, which were crucial for successful operations. Seven ONH cases , 24, 30 Based on the location at optic pathway and the MRI performances, optic pathway glioma (OPG) and optic nerve sheath meningioma (ONSM) are two commonly considered differential diagnoses of ONH. Here, we summarized the key points in the differential diagnosis based on abovementioned neuroimaging features Table 2.The OPG is a kind of low-grade glioma which occurs majorly in children. Histologically, most OPGs are classified as PA. PA typically appears as fusiform enlargement or uniform thickening of optic nerve . It has There are three major points that may contribute to the differentiation of OPG and ONH: (1) flow voids as the most specific marker of ONH are rarely seen in OPG; (2) prominent peritumoral edema is also suggestive of ONH. The OPG, like most low-grade tumors, induces no or little peritumoral edema ; (3) theONSM is a rare benign tumor originating from meningoepithelial cap cells of arachnoid villi. More than 90% ONSM are intraorbital. While it has a similar intensity pattern to ONH on MRI, ONSM usually appears as tubular or globular enlargement of optic nerve . Cystic Optic nerve hemangioblastoma is extremely rare. Including this case, 40 cases have been reported since 1940. Despite the fact that MRI is the major diagnostic tool of ONH, few of these case reports provided detailed descriptions of MRI results for reference in clinical practice. On the other hand, ONH has similar MRI performances to OPG and ONSM, making it highly challenging to accurately diagnose ONH. Therefore, more experience on MRI imaging is needed to improve the diagnosis. In this study, we reported a 41-year-old male with recurrent hemangioblastoma in both cerebellum and optic nerve. The manifestations on conventional MRI were described in detail. Moreover, advanced MR techniques were employed for the first time to explore for more diagnostic tools for ONH. The results revealed that the mass was evidently hyperintense on ADC maps but showed no abnormity on MRA, which was out of expectation.We also reviewed all present case reports to summarize the MRI and angiography results of ONH and discussed the differential points between ONH and easily confused diseases. Similar to cerebellar hemangioblastoma, most ONH cases possess iso- or hypointensity on T1WI and hyperintensity on T2WI. The prominent enhancement and well-defined boundaries are prevalent. In addition, structural features including flow voids and extensive peritumoral edema are also widely accepted as suggestive evidences of ONH. The flow void effect has high specificity but is not sensitive enough. Larger masses seem to be more likely to cause the effect. Extensive peritumoral edema is a unique property of ONH absent in cerebellar hemangioblastoma. Optic pathway is often involved due to its low resistance environment and contralateral vision can be impaired consequently, indicating the necessity of timely and complete resection. It is also found that the solid-form hemangioblastoma accounts for the vast majority of ONH, instead of cystic form which typically presents in cerebellar hemangioblastoma. This possibly resulted from drainage of transudate through the extensive edema. Therefore, the absence of typical large cysts with mural nodules should not be recognized as a clue for excluding ONH during diagnostic process.ONH is difficult to be distinguished from meningioma and glioma with simply conventional imaging approaches, especially when typical clinical features mentioned above are not observed. Though few studies and reports focus on applying advanced MRI techniques on ONH, experience from cerebellar hemangioblastoma can also be informative. It has been reported that ADC values in hemangioblastoma are distinctively higher when compared with most other posterior fossa tumors such as meningioma and metastases, indicating its potential diagnostic value in ONH. We here applied ADC on the evaluation of ONH for the first time and observed a hyperintense mass, similar to the manifestations of cerebellar hemangioblastoma.In addition, high rCBV ratio on PWI can be another exclusive marker of hemangioblastoma. Cerebellar hemangioblastoma shows a remarkably higher rCBV ratio than metastases, meningioma, and both low-grade and high-grade gliomas, and thus can be easily distinguished. However, PWI results in ONH have not been reported yet.Angiography used to be crucial for the diagnosis of ONH before MRI was advanced. Almost all cases performing angiogram revealed hypervascular masses originating from ophthalmic arteries. In contrast, MRA has not been frequently used in either optic nerve or cerebellar hemangioblastoma.In conclusion, various MR modalities including DWI, ADC, and PWI provide new insights to the imaging manifestations of ONH. For diagnosed VHL patients, an optic nerve lesion is highly suspected to be hemangioblastoma. However, for patients who do not meet the clinical diagnosis criteria of VHL, any possibility of the lesion should be carefully evaluated. In such cases, preoperative diagnosis by advanced MRI techniques can be decisive for their following examinations, treatment, and prognosis. Though more ONH cases employing these advanced techniques are needed to systematically review their diagnostic performances, these tools are valuable auxiliary approaches that can prompt the differentiation of ONH in these situations.The original contributions presented in the study are included in the article/Written informed consent was obtained from the individual(s) for the publication of any potentially identifiable images or data included in this article.MD composed the manuscript and reviewed literature. LY provided figures and reviewed literature. JK and RW contributed to the organization and revision of the manuscript. HY reviewed the MRI images in case reports, analyzed the results, and edited the manuscript. MF selected and managed the patient case and edited the manuscript. All authors contributed to the article and approved the submitted version.This work was supported by Chinese Academy of Medical Sciences / Peking Union Medical College Postgraduate Teaching Innovation Fund (No. 10023201900107).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.


We present a case of cutaneous botryomycosis of the lower leg in a young adult male. Botryomycosis is a chronic granulomatous response to bacterial infection. As a cutaneous lesion, it can easily be mistaken for a malignant, autoimmune or inflammatory mass. We were able to successfully treat our patient with primary surgical excision, vacuum-assisted closure (V.A.C.™) dressing and subsequent split thickness skin graft (STSG). Use of a V.A.C. dressing with subsequent grafting has not previously been reported in the literature.  We report a case of cutaneous botryomycosis of the leg in a 37-year-old Caucasian male treated with primary surgical excision, vacuum-assisted closure  and subsequent split-thickness skin grafting (STSG). Botryomycosis is a disease of chronic inflammatory lesions caused by a granulomatous response to bacterial infection . The lesWith only 200 reported cases in the English literature, botryomycosis is a rare entity. There are no previously reported cases of surgical excision with subsequent use of a V.A.C. dressing and skin grafting and we believe the case presented below shows that this is a suitable reconstructive option. Staphylococcus aureus (MRSA) and Enterococcus. Our patient was a 37-year-old Caucasian male who presented to the emergency department with a 30-day history of a necrotic, ulcerating right calf lesion. He was systemically well and did not have any pain. He denied any preceding trauma or inciting triggers. He had been treated by his general practitioner with oral amoxicillin/clavulanic acid, however, the lesion failed to improve. His past medical history included alcoholism, anxiety disorder, recurrent lower limb cellulitis and a right ankle fracture requiring internal fixation. He was an active smoker with no history of diabetes mellitus. On examination, the 5cm diameter lesion had areas of ulceration and necrosis with surrounding cellulitis Figure . There wThe patient underwent primary surgical excision of the lesion. Macroscopically, there was low-grade inflammation which is why we delayed grafting. A V.A.C. was applied with negative pressure at 125mmHg with continuous suction. Histopathology with Periodic acid-Schiff staining revealed extensive suppurative necrosis with fibrosis. The base of the lesion contained granules with blue-stained bacterial organisms surrounded by an intense eosinophilic coat. This finding is in keeping with “Splendore-Hoeppli Phenomenon”, a finding characteristic of botryomycosis. There was no evidence of malignancy and no fungal organisms were identified. He was treated with 48 hours of intra-venous clindamycin and discharged with a two-week course of oral clindamycin as guided by the infectious diseases team. The V.A.C. was managed by community nurses. Upon review in the clinic, the wound bed appeared healthy with good granulation tissue. The cellulitis had improved Figure . The patient’s V.A.C. dressing was only attended to in clinic due to anxiety surrounding changing these dressings at home. Once the inflammation settled and the wound base appeared healthy, the patient was re-admitted and underwent a split-thickness skin grafting procedure. Upon follow-up, the graft took well Figure .Botryomycosis may be either cutaneous (75% of cases) or visceral, typically affecting the lungs but possibly involving the liver, spleen, brain or kidneys . Its namStaphylococcus aureus (40% of cases) followed by Pseudomonas aeruginosa (20% of cases) and Escherichia coli. Other implicated organisms include Proteus and Streptococcus species [The most frequently implicated organism is Botryomycosis is thought to arise when there is a balance between the virulence of the involved bacteria, the size of inoculum and an impaired host response. Too little inoculum, low bacterial virulence and a robust host response favours clearance of the infection. In the opposite scenario, bacterial infection overwhelms the host response forming an abscess . MicroscThere are reported cases of successful management with antibiotics exclusively, however combination with surgical debridement is often used ,8. In ouIn conclusion, this rare clinical entity may easily be mistaken for a malignant lesion, or a non-infective inflammatory or autoimmune mass. In cases where lesions are smaller and not yet necrotic, primary antibiotic therapy would be feasible. However, larger lesions will most likely be non-responsive due to a lack of antibiotic penetrance and the presence of necrotic tissue requiring debridement. Our case highlights that surgical excision and V.A.C. dressing allow time to treat the infection. An STSG is a suitable reconstructive option.


Rhabdomyosarcoma (RMS) is the most common soft-tissue tumor in children, frequently affecting the nose, nasopharynx, and paranasal sinuses. RMS of this sinonasal region manifests with non-specific nasal symptoms of allergic rhinitis, sinusitis, or upper airway infection. Associated ocular symptoms are rare. We describe a young girl who presented with acute onset squint secondary to sinonasal RMS. Imaging showed an aggressive sinonasal tumor with oropharyngeal, intraorbital, and intracranial extension. Histopathological findings were consistent with a diagnosis of rhabdomyosarcoma, embryonal type. The patient deteriorated rapidly due to disease complications. We provide a literature review of pediatric sinonasal RMS with various manifestations. Rhabdomyosarcoma (RMS) is a malignant tumor featuring skeletal muscle differentiation. It is the most common soft tissue sarcoma among children. However, it is still rare, accounting for less than 5% of all paediatric tumors . Most RMA 4-year-old girl was referred from an otorhinolaryngology (ORL) clinic for eye assessment. She had sudden onset left divergent squint for one month followed by progressive blurry vision and painless proptosis. The right eye developed almost the same symptom two weeks later. The parents reported that the girl had recurrent epistaxis a few days prior to this visit. The patient was otherwise previously healthy and active. There was no history of a blocked nose or rhinorrhea; no diplopia, headache, or vomiting episodes were reported. Past medical history was unremarkable.Examination of the left eye revealed a marked non-axial proptosis with lagophthalmos, relative afferent pupillary defect, and total ophthalmoplegia. Slit-lamp examination showed a generalised conjunctival injection, exposure keratopathy with inferior paracentral epithelial defect. The right eye had mild proptosis with complete paralysis of abduction; other findings were unremarkable. Fundus examination revealed papilledema with choroidal folds noted on the left eye. Besides ocular findings, the patient also had fullness and swelling of the left cheek. The nose was swollen, slightly distorted with the presence of a hemorrhagic fungating mass in the left nostril. Multiple cervical lymphadenopathies were present. An urgent computed tomography (CT) scan of the brain, orbit, and paranasal showed an aggressive left sinonasal enhancing mass with intracranial extension superiorly involving bilateral cavernous sinus, bifrontal extradural space, and the left temporal lobe. The tumor also extended to the bilateral orbit, left infratemporal fossa with involvement of left pterygoid muscle, posteriorly occupying the whole nasopharynx and inferiorly to the oropharyngeal region Figure . A block of friable nasal mass was detached during a forceful sneezing episode in the ward, prior to the planned biopsy. It was sent for definitive histopathological analysis. The microscopic and immunohistochemical findings were consistent with a diagnosis of rhabdomyosarcoma, embryonal type Figure . Both parents were counselled regarding tumor progression and the potential complications. The tumor growth was aggressive and had already advanced to neighboring structures. Thus, tumor radioablation followed with chemotherapy was the best option. However, they decided for alternative traditional treatment. The patient was discharged against medical advice due to parents’reluctance for medical and surgical treatment. About one month after discharge, the parents brought the patient back to the hospital in a severely ill condition. Parents reported that child deteriorated at home, was unable to tolerate neither feeding nor fluids, and was bed-bound ever since. There was severe disfiguration of the face with midfacial fullness, and a huge bulk of necrotic tumor mass protruded out from the nose Figure . The patSinonasal RMS usually present with symptoms mimicking allergic rhinosinusitis. It is essential to conduct a detailed examination followed with biopsy and histopathological analysis to confirm the diagnosis. A recent epidemiological and end result study has defined six subtypes of RMS, which include embryonal, alveolar, pleomorphic, spindle cell, mixed type, and ganglionic differentiation type . A signi There was one case in which the parents denied any ocular symptoms but noted the patient to have mild proptosis during the initial assessment [We reviewed nine cases of confirmed sinonasal RMS from 2003 until 2017. The pleomorphic subtype was seen only in one case, whilst the rest were embryonal -10. We ssessment . Our patFive of the previously reported cases were successfully treated and survived, whereas another two died despite treatment given due to disease progression and complication. Six cases underwent tumor resection via endoscopic approach, followed by chemoradiotherapy or chemotherapy alone. The patient with pleomorphic subtype who already had intracranial extension successfully went into remission after combination treatment of chemoradiotherapy, without any tumor resection . ChemothLike other childhood cancers, mortality is the main concern. High-risk groups with poor prognostic factors include children with parameningeal localization, larger tumor size, younger age at presentation, and distant metastasis ,12. Chil Despite being mostly fatal, with great care, RMS is curable. It is crucial for paediatricians, oncologists, and radiologists to work together to decide the best option of treatment. Most important is providing clear advice and continuous support to the parents. Multidisciplinary care and multimodality approach composed of aggressive chemotherapy, radiotherapy, and tumor debulking have shown to induce remission, improve survival, thus reducing morbidity and mortality . DespiteAn abrupt onset of squint always require urgent evaluation and prompt referral to ophthalmologists, regardless of the age. It could be the first presenting symptom of a life-threatening condition, such as advanced agressive tumor in our case. The survival rates for patients with embryonal RMS are generally the highest, but tumor size, localisation, grade, and presence of metastatic disease were more predictive of survival. Whether primary or secondary, sinonasal RMS generally has a poorer prognosis due to adjacent parameningeal localisation.


Physical examination of the abdomen revealed a giant non-tender mass in the right quadrant. The patient was transfused with 4 units of blood. Abdomen computed tomography (CT) scan revealed a retroperitoneal mass of 20cm with calcification involving the liver, the kidney and surrounding the vena cava. He was referred to a multi-disciplinary team (MDT) to perform a CT scan fine-needle-aspiration (FNA) with a differential diagnosis of lymphoma, sarcoma or renal neoplasia.Retroperitoneal tumors give non-specific symptoms until they have reached a substantial size. Hystopathological types are: sarcomas such as liposarcoma (70%) and leiomyosarcoma (15%). Other retroperitoneal neoplasms include primary lymphoproliferative tumours (Hodgkin's and non-Hodgkin lymphoma) and epithelial tumours  or metastatic disease. A 57-year-old male was referred to the emergency department with acute onset of severe melena, right back pain and lipothymia. He had no medical history. On physical examination arterial blood pressure was 70/60mmHg, pulse rate was 100b/min, oxygen saturation of 90%. Blood exams demonstrated lymphocytes 61 x 10


To report a rare case of primary orbital pleomorphic liposarcoma and present a relevant literature review.An 11-year-old boy presented with an enlarging, painless lower right eyelid mass that was noted 4 months ago. Imaging revealed a 3.2 × 2.1 × 3.7-cm-sized well-circumscribed lobulated mass. Biopsy revealed a pleomorphic spindle cell neoplasm that was consistent with a pleomorphic liposarcoma. A systemic evaluation found no evidence of distant metastasis. Despite four chemotherapy cycles, the mass size increased at follow-up. Total orbital exenteration was performed 1 year after confirming a diagnosis of pleomorphic liposarcoma. At the 6-month follow-up, the patient was alive with no signs of recurrence.Pediatric pleomorphic liposarcoma is a very rare entity. Its definite diagnosis relies on histopathological results. In the absence of systemic metastasis, total orbital exenteration is the optimal approach for local control. Only seven case reports of primary orbital pleomorphic liposarcoma have been found in the literature since 1990, and only two of them were in patients aged <20 years.2An 11-year-old Asian boy with an enlarging lower right eyelid mass was referred to our hospital for further evaluation and complete tumor removal. The child had homozygous hemoglobin E disease, with no anemic symptoms, as an underlying disease. He denied any significant past ocular history or family history of malignancy at a young age. Four months ago, he first noticed this painless mass of approximately 1 cm. Two months after the mass had developed, the child underwent contrast-enhanced orbital magnetic resonance imaging (MRI), which revealed a 3.2× 2.1 × 3.7-cm-sized well-circumscribed lobulated mass on the lower right eyelid, abutting on the right inferior rectus muscle and causing superior right eye globe displacement.On physical examination, his best-corrected visual acuity was 2/60 OD and 6/12 OS, with normal pupil reaction. External examination showed a 5-cm- painless, firm lower right eyelid mass. The mass caused superior globe displacement, complete ptosis, marked conjunctival chemosis, and limited extraocular muscle movement, with the greatest restriction on the inferior directions A. The leNeoadjuvant chemotherapy , including ifosfamide and doxorubicin, was administered as four cycles in total (3 weeks apart). The mass slightly increased in size at the follow-up examination, and a trace relative afferent pupillary defect of the right eye was noted B. Orbita3Liposarcoma of the head and neck region is a rare entity. The pleomorphic subtype is a high-grade undifferentiated sarcoma with a variable number of pleomorphic lipoblasts and has the most aggressive course, with a 1-year survival rate of approximately 45%.The tumor was surgically resected in all of the cases, either via local resection or exenteration. Two patients died, whereas the others showed no sign of recurrence or were lost to follow-up.12Liposarcoma is extremely uncommon in children. The most common primary site in adults is the extremities. Madge et al. reported on a 19-year-old Caucasian man with primary orbital pleomorphic liposarcoma.The treatment option for liposarcomas is primarily surgical resection, and it is generally recommended to excise at least 2 cm of negative surgical margins in cases of soft tissue sarcomas.4We described the clinical features of an 11-year-old boy with primary orbital pleomorphic liposarcoma, a rare entity. Diagnosis primarily depended on histopathological findings obtained via biopsy. In the absence of systemic metastasis, total exenteration of the orbit with adequate surgical margin seemed to be the best treatment option. Adjunctive chemotherapy or radiotherapy might improve local control, although the decision should be weighed against the risk. Clinicians should consider liposarcoma as a differential diagnosis of orbital masses in children.Trakanta Wannapanich: Conceptualization, Methodology, Validation, Writing - Original Draft, Writing - Review & Editing, Visualization.Paitoon Pratipanawat: Validation, Writing - Review & Editing, Supervision, Project administration, Funding acquisition.Both patient and patient's legal guardian consented to publication of the case in writing. The patient and his parents have given their permission to publish their clinical data and images.Faculty of Medicine, Khon Kaen University, Thailand.The authors declare that there are no conflict of interests of this paper.


Introduction: Meckel’s diverticulum (MD), a remnant of the omphaloenteric duct, is among the most frequent intestinal malformations. Another embryonic vestige is the urachus, which obliterates, becoming the median umbilical ligament; the failure of this process can lead to a urachal cyst formation. We present a case of Meckel diverticulitis misdiagnosed as an infected urachal cyst. Presentation of case: A 16-year-old girl presented with hypogastric pain, fever and vomiting. She had undergone an appendectomy 6 years prior and no digestive malformation had been documented. In the last 2 years, she had 3 events of urinary tract infections with Escherichia coli, and anabdominal ultrasound discovered a 28/21 mm hypoechogenic preperitoneal round tumor, anterosuperior to the bladder. We established the diagnosis of an infected urachal cyst, confirmed later by magnetic resonance imaging. Intraoperative, we found MD with necrotic diverticulitis attached to the bladder dome. Discussions: Meckel’s diverticulum and urachal cyst (UC) are embryonic remnants. Both conditions are usually asymptomatic, being incidentally discovered during imaging or surgery performed for other abdominal pathology. Imaging diagnosis is accurate for UC, but for MD they are low sensitivity and specificity. For UC treatment, there is a tendency to follow an algorithm related to age and symptoms, but there is no general consensus on whether to perform a routine resection of incidentally discovered MD. Conclusion: Preoperatory diagnosis of MD represents a challenge. We want to emphasize the necessity of a thorough inspection of the small bowel during all abdominal surgical interventions and MD surgical excision regardless of its macroscopic appearance. These two actions seem to be the best prophylaxis measures for MD complications and consequently to avoid emergency surgery, in which case more extensive surgical procedures on an unstable patient may be needed. Both UC and MD are embryologic vestiges originating in the yolk sac (endoderm). The umbilical cord contains various elements such as the allantois and the omphaloenteric duct. The allantois obliterates, forming the urachus, while the omphaloenteric duct resorbs completely ,2. If thRegarding the imaging diagnosis methods, UC can be found on an abdominal ultrasound (US), but in MD cases, the correct diagnosis is provided by laparotomy or laparoscopy ,4,5. BecWe report a challenging case of a 16-year-old girl, presenting with symptoms and imaging investigations matching with an infected UC, but during surgical intervention, we found Meckel diverticulitis attached to the bladder.A 16-year-old girl showed up at a general practitioner with a fever (39 °C), median subumbilical pain and dysuria. In the last 2 years, she had experienced 3 events of urinary tract infection (UTI) with Escherichia coli. Associated reno-urethral malformations were not documented during an US. The recommendation was to start treatment with ciprofloxacin and ibuprofen. Because her fever and pain persisted after 5 days, she was referred to our hospital. She complained of hypogastric pain with fever (39 °C), nausea and vomiting. She had been operated on 6 years prior for acute appendicitis (open surgery) and no digestive malformation was documented. The menarche was at the age of 14 years, but during the last 6months, she had experienced menstrual irregularity  and dysmenorrhea. Affirmatively she had no sexual activity. The physical examination revealed hypogastric tenderness without peritoneal signs, with median subumbilical warmth and erythema, but no drainage from the umbilicus was ever seen. Laboratory investigations found a high level of CRP (11.60 mg/dl), and urinalysisrevealedproteinuria  and many epithelial cells, but the urine culture was sterile. The Addis–Hamburger test showed high levels of red and white blood cells and the rapid test for the detection of fecal occult blood in stool was positive.The abdominal ultrasound (US) discovered a 28/21 mm hypoechogenic round tumor in the midline of the hypogastric region, anterosuperior to the bladder and posterior to the abdominal wall, with diffuse mural thickening. The uterus and both ovaries appeared to be normal. We decided to perform a pelvic MRI which showed a 16/18/32 mm inhomogeneous nodular lesion, apparently on the urachal tract; the bladder was in repletion, with mural thickening of the anterosuperior wall. The left ovary presented a 41/34 mm cyst, considered as a hemorrhagic corpus luteum .Because the radiologists requested additional investigations, an abdominal–pelvic CT scan with intravenous contrast was performed, adding an intense contrast capture in the periphery of the lesion with edema of the prevesical space .The diagnosis of an infected urachal cyst was established and we decided to manage the condition with a second generation of cephalosporins and observation. After 24 h, both symptoms and local inflammatory signs improved and 1 week later she was discharged from the hospital. After 10 days, the patient presented at the Emergency Department with signs of intestinal occlusion: infraumbilical pain, abdominal meteorism and vomiting. A new abdominal US was performed which showed the same cystic tumor as before, but between its anterosuperior pole and the prevesical fat there was a canal-like structure 28 mm long and 7 mm diameter, which was supposed to be the small intestine adherent to the infected UC.We decided on an open-surgery intervention via a median infraumbilical laparotomy. The rectus abdominis muscles and properitoneal fat were edematous. In the abdominal cavity, we found a 6 cm long and 2.5 cm wide case of MD covered by the omentum and adherent to the midline of the anterior abdominal wall. Between the necrotic tip of the MD and the bladder, there was a little abscess (the antibiogram showed the presence of Staphylococcus epidermidis). Because the inflammatory process had damaged the corresponding detrusor muscle, we resected that area, and the bladder was closed using 3-0 vicryl. For the removal of MD, we performed a segmental resectionanastomosis. Both ovaries were normal but the uterus was large, with thick walls. We placed a urinary catheter which was removed after 3 days. Pathology revealed acute inflammatory tissue without malignant or ectopic mucosal cells. Postoperative, the recovery was uneventful and the patient was discharged after 8 days. Three months later she had no more digestive or urinary symptoms and experienced her first normal menstrual cycle.The allantois is a caudal diverticulum of the yolksac. Around the fifth gestational month, it will be completely obliterated, becoming the median umbilical ligament located in the prevesical space of Retzius ,2. If thUC is ordinarily small and silent, but bacterial contamination or epithelial degeneration can enlarge the lumen and cause symptoms . In thisRegarding the UC treatment, there is a tendency to follow an algorithm related to age and symptoms. Patients under 1 year of age, even symptomatic, are treated medically under US surveydue to frequent spontaneous resolution; surgery is reserved for recurrent infections. For older children or adults, the treatment of choice for symptomatic UC is surgical excision because of the high rate of malignant degeneration. So far, surgical intervention has been open excision, but lately, the laparoscopic approach is preferred ,4,8.The omphaloenteric duct joins the midgut lumen to the yolk sac. It gradually obliterates and later resorbs completely. If the process fails it can causemany anomalies such as MD, an umbilical cyst, omphalomesenteric fistula or umbilical sinus . MD reprComplementary imaging studies are of little value; abdominal ultrasound, contrast-enhanced CT scan, angiography ormagnetic resonance imaginghave low sensitivity and specificity, often producing false-negative or false-positive results. The most useful investigation for MD detection in children is considered to be the technetium-99m pertechnetate scan, with a sensitivity of 80–90% and specificity of 95%, but only if the case of MD has ectopic gastric mucosa. In fact, exploratory laparotomy or laparoscopy are the investigations that can establish a correct diagnosis ,5.Regarding treatment, there is no general consensus on whether to perform a routine resection of incidentally discovered MD . HoweverThree procedures have been described: segmental (‘‘T’’-shaped) resectionanastomosis, wedge resection and tangential stapling; for the last two options there remains a small risk of leaving residual heterotopic tissues at the MD base .In our case, we excluded MD pathology because the patient had been operated on for acute appendicitis and we supposed that the mandatory intestinal exploration certified its absence. Moreover, the symptoms were specific for an infected UC . The clinical diagnosis was sustained by imaging investigations and consequently, we decided to adopt a conservative attitude, treating the infection with antibiotics. Even a week later when the patient returned with clinical signs of intestinal occlusion and the abdominal US showed the cystic tumor attached to a canal-like structure supposed to be the small intestine, we thought that the infected UC was the cause of the symptoms because of its inflammatory adhesions to the bowel. We were extremely surprised when we found a MD abscess with an important detrusor muscle destruction.We searched in the literature and we discovered only one pediatric case of Meckel’s diverticulitis presented with periumbilical cellulitis due to its attachment to the anterior abdominal wall .Because of its various clinical presentations and despite high-performance imaging investigations, MD diagnosis is still a challenge. The purpose of this case report is to remind the importance and necessity of a thorough inspection of the last 150 cm of the ileum whenever an abdominal intervention is performed, as well as to recommend MD surgical excision regardless of its macroscopic appearance. These two actions seem to be the best prophylaxis measures for MD complications and to consequently avoid emergency surgery, in which case more extensive surgical procedures on an unstable patient may be needed.


Glomus tumors are rare and usually benign. The malignant form (glomangiosarcoma) comprises <1% of all glomus tumors. There are limited reports that describe glomus tumors in the nasal cavity. However, to the best of our knowledge, glomangiosarcoma of the nasal cavity was never reported in humans. We report on the first case of nasal cavity glomangiosarcoma in a 59-year-old male who presented with a bleeding mass in his right nostril. We completely excised the lesion with a 0.7-mm free margin, and the histopathologic examination was consistent with glomangiosarcoma. A 6-month follow-up illustrated no evidence of recurrence or distant metastasis. Although it is rare, glomus tumors should be in the differential diagnosis of nasal cavity tumors. Histopathologic examination is essential for glomangiosarcoma diagnosis. Treatment requires complete excision with free margin, alongside careful clinical and radiological follow-up. Glomus tumor is mesenchymal neoplasm originating from the glomus body, modified arteriovenous shunts involved in thermoregulation; they are rare neoplasms, mostly found in the older population with a higher incidence in females , 2. It uA 59-year-old male with a history of intermittent epistaxis of 20-day duration presented with active bleeding from his right nostril, which was refractory to compression; we applied nasal packing with adrenaline, with only minimal improvement. Physical examination revealed a red-purple mass lesion obstructing the right nasal passage. A computed tomography scan showed a mass in the right nasal cavity attached to the septum measuring 1 × 1.5 cm, shown in Histopathologic examination of the resected mass showed proliferation of atypical spindle cells with atypical mitotic figures, focal areas of necrosis, marked nuclear atypia and pleomorphism . ImmunohOn postoperative follow-up, the patient had no complications and was improving. He had magnetic resonance imaging 3-week post-surgery, which showed a faint enhancing area of 5 mm seen in the right nasal cavity, likely representing a focal area of post-surgical changes. There was no evidence of any lesions. The patient has had a positron emission tomography (PET) scan 2 weeks later, which demonstrated no signs of distant metastasis. Still, there was active, hypermetabolic soft tissue density in the right nasal cavity; this probably was a benign post-surgical finding. Yet, we could not exclude malignant growth. We performed another surgical intervention through a midface degloving approach including resection of the suspicious lesion along with ~0.7-mm grossly free margin, besides taking biopsies from the margins and resection of the cartilage preserving the mucosa on the other side.Histopathologic evaluation of the resected lesion revealed no evidence of residual disease and exhibited a picture of chronic inflammation and no signs of malignancy. We performed a PET scan 6 months later, and it showed no signs of active lesions or distant metastasis.Most glomus tumors are small neoplasms that are benign, usually arise in the subungual zones of the digits where glomus bodies are most abundant, but they still can originate in unusual sites such as deep soft tissues, gastrointestinal tract, respiratory tract and rarely in the head and neck . GlomangGlomus tumors of the nasal cavity mainly manifested with epistaxis and obstructive symptoms. Yet, patients with localized pain and asymptomatic cases were reported . Our patThe clinical symptoms with radiologic findings are usually insufficient to reach a definite diagnosis. However, hematoxylin and eosin staining, coupled with immunohistochemical staining, is required to achieve the definitive diagnosis of glomangiosarcoma .Histologically, the classic features of glomus tumors are uniform sheets of small, round cells with round to oval nuclei . The tumGlomus tumors have a broad differential diagnosis. When they arise in the nasal cavity, we should distinguish them from other tumors, such as hemangiopericytoma and angiosarcoma. The distinction between hemangiopericytoma and glomus tumors is based on the expression of smooth muscles features found in glomus tumors, such as positive staining for SMA, whereas hemangiopericytomas lack these features . Angiosaet al. classified glomus tumors with atypical features into glomangiosarcoma, a malignant glomus tumor; symplastic glomus tumor presents with nuclear atypia alone; glomangiomatosis a glomus tumor with diffuse growth and benign histological features and glomus tumor of uncertain malignant potential.Folpe They suggested considering glomangiosarcoma in lesions that meet at least one of the following criteria, deep-sited location with a size >2-cm, atypical mitotic figures, or the combination of moderate to high nuclear grade and mitotic activity (5 mitoses/50 high-power field (HPF)). They found the metastasis rate in glomangiosarcoma cases can be as high as 40% [et al., criteria for diagnosing glomangiosarcoma as the tumor displayed atypical mitotic figures; these, along with the presence of focal necrosis, nuclear atypia and pleomorphism, are alarming features for high metastatic risk.Our patient’s tumor was consistent with Folpe We approached the mass initially by complete excision followed by histopathologic evaluation. Then, we resected the remnant hypermetabolic lesion with a 0.7-mm free margin without adjuvant chemotherapy or radiotherapy, besides close clinical and radiologic follow-up. The patient had no local recurrence or distant metastasis after a 6-month follow-up. Based on our experience, complete excision of the mass with macroscopic free margins is curative. However, glomangiosarcoma has a fair metastatic and recurrence rate. Therefore, clinical and radiological follow-up for such patients is crucial.


Most studies on pigmented villonodular synovitis (PVNS) of the temporomandibular joint (TMJ) with skull base extension mostly are case report. Here, we summarize the clinical features, treatments, and outcomes of PVNS of the TMJ with skull base extension in a large case series. We reviewed the clinical information relating to patients diagnosed with PVNS of the TMJ with skull base extension information of patients in our center between 2011 and 2020. We reviewed 10 patients . All cases had presented with a unilateral lesion extending the middle skull base. PVNS of the TMJ with skull base extension occurred on the left side in 6 patients (60%) and on the right side in 4 patients (40%). Of the 10 patients, pain and mass were the most prevalent symptoms. All patients received surgery and no recurrence was seen after 35.90 ± 25.35 months follow-up. Despite destructive biological behavior, surgery can achieve an excellent outcome for patients with PVNS of the TMJ with skull base extension. An en bloc resection may prevent recurrence and provide long-term relief. Radiotherapy may be reserved for subtotal excision and recurrent lesions but require further investigation. This condition often affects larger joints such as the knee and hip, although other joints may also be affected, such as the ankles and shoulders2. Initially considered as an inflammatory disease, cytogenetic studies revealed that PVNS is associated with a chromosomal translocation: t   that fuses COL6A3 (encoding collagen type VI α3) to colony-stimulating factor 1 (CSF-1), and also revealed that PVNS is a neoplasm that can exhibit destructive behavior3. The main treatment is resection which can achieve excellent clinical outcomes. Studies have also shown that this disease may be caused by chronic inflammation4, although recurrent trauma and hemarthrosis may also be risk factors5.Pigmented villonodular synovitis (PVNS), also referred to as a diffuse-type tenosynovial giant cell tumor (D-TGCT), or a giant cell tumor (TGCT) of the tendon sheath, is a rare, proliferative, locally destructive, and mesenchymal neoplasm with a relatively higher recurrence ratePVNS in the TMJ is extremely rare; the combination of this condition with the extension of the skull base is even rarer. In this present, we reviewed a large series of cases from our hospital to summarize this condition by its clinical features, treatment, and outcomes.The institutional review board of West China Hospital of Sichuan University approved the present study in accordance with the World Medical Association Declaration of Helsinki (2002 version). Written informed consent was obtained from each patient or family member. Patient information was identified by different terminologies, including PVNS, TGCT, and giant cell tumor of the tendon sheath in the TMJ with a skull-extending base. We retrospectively reviewed 10 patients from January 1, 2020, to December 31, 2020. The clinical information, including age, sex, complaint, radiological features, pathological findings, treatment methods, and clinical outcome were recorded.Preoperative and postoperative contrast-enhanced magnetic resonance imaging (MRI) were scheduled in all patients. The latter was given to evaluate the surgical extent of the tumor. Gross total resection (GTR) was defined that no residual tumor was observed on postoperative MRI, otherwise, subtotal resection (SR) was considered. Patients were performed with hematoxylin–eosin stain (H&E) and immunohistochemical examination. The follow-up interval was 3 months, 6 months, and 1 year after surgery. Contrast enhancement MRI was performed during follow-up. Telephone consultation was constant.All experimental protocols were approved by Ethics Committee on Biomedical Research, West China Hospital of Sichuan University. All methods were carried out in accordance with the relevant guidelines and regulations. Informed consent was obtained from the participant.The characteristics of 10 patients were shown in Table The lesion on MRI presented expansive growth and slight enhancement with skull base invasion associated mass effect on the temporal lobe Fig. A,B. EighMicroscopically, the H&E findings were presented. The lesion revealed clusters of multinucleated giant cells and adjacent hemosiderin-laden mononuclear cells Fig. A,B.FigurAll patients received surgical treatment with the preauricular infratemporal fossa approach Fig. . Nine paPVNS of the TMJ with skull base extension is exceedingly rare. Almost all researches were a case series report and retrospective study. We aimed to present a large case series of the condition.6. The disease usually occurred in the middle-aged, although it could be seen even at the age of 18 in our case series. Pain and mass were the most common presentations; symptoms related to neurological symptoms such as hearing loss and tinnitus were not uncommon, especially in cases with serious petrosal bone erosion7. Trauma has been reported to be contributing factor5, but it was not common.Contrary to other studies, our study found that the condition had male predominance (1.64:1) which was contrary to other studies8. Careful reading images before surgery are important for surgical planning. On MRI, the lesion generally shows low signal intensity on T2 images and “blooming” artifact on gradient-echo images due to the high hemosiderin concentration9. The lesion on contrast MRI presents variable levels of enhancement. CT mainly presents bony destruction of the mandibular condyles and skull base invasion and areas of central calcification. The differential diagnosis mainly consists of chondroblastoma, reparative granuloma, chondrosarcoma, giant cell tumor of bone, and synovial sarcoma. Digital subtraction angiography may show highly vascular lesions with multiple arteries blood supply and arteriovenous shunting, especially for giant tumors. It is useful to conduct preoperative embolization to decrease blood loss during operation. One of our cases was performed digital subtraction angiography embolism because the patient had a big tumor with 3.8 cm in diameters.Imaging examinations are necessary for the initial and differential diagnosis, and surgical evaluation10. Pathologic results reveal that the lesion is rich in giant cells and focal cartilage formation. Different kinds of cells, including mononuclear histiocytes, multinucleated giant cells, and xanthoma cells, are present in the lesion11. The differential diagnosis includes giant cell granuloma, giant cell tumor, and brown tumor12. Due to the overlapping of histopathological characteristics of these lesions, the definitive diagnosis may need additional testing tools. A brown tumor can be ruled out with normal parathyroid and calcium levels. Giant cell tumors are generally more destructive. Giant cell granulomas are more commonly bone-based. Evidence of hemosiderin deposition may also aid in the appropriate diagnosis of PVNS6. In our cases, one patient has undergone an FNAB test.Pathologic evaluation is essential for a definitive diagnosis. Biopsy and postoperative pathological examination are the most common method. Because PVNS of TMJ is involved in the facial region and the tumor is relatively superficial, the pathologic examination before surgery provides valuable information for diagnosis and treatment. FNAB with the guidance of ultrasound or CT is a simple method13. RT after SR for residual tumors is an adjuvant treatment14. The most common radiation dose is 35–60 Gy in 20–30 fractions. Therefore, GTR should be conducted when it was feasible. When complete removal of the tumor could not be achieved, RT after surgery is recommended. In our series, GTR was achieved in 9 patients and 1 patient had SR for facial nerve preservation. Truly GTR included completely bony resection and soft tissue resection. We often perform extended removal for suspicious bony invasion including the greater sphenoid wing, squamous and petrous parts of the temporal bone. All our cases were extradural lesions, invasion of the periosteal layer of the dura mater was found in 10 patients. Meticulously sharp separation was performed to remove the outer layer dura for complete resection and preserve the inner layer to prevent leakage of CSF. The operation involved multi-disciplinary cooperation, including neurosurgeons, otolaryngologists, and maxillofacial surgeons. Multi-disciplinary cooperative surgeries could achieve more radical resection, reliable skull base reconstruction, and better cosmetic results.The most effective treatment for the condition is surgery. GTR leads to a lower recurrence rate and is associated with a longer period of relief when compared with SR. However, GTR may bring damage to occlusal function because of the destruction of the functions of the TMJ. Some scholars think that SR may be a risk factor for recurrence6 believed that it was not necessary to reconstruct the middle skull base and that autologous fat can be used to fill large defects. However, Liu et al.15 reported that repairing bony defects in the skull base can provide a satisfying contour and stable support for the brain. Unfortunately, there is a lack of enough information relating to the comparison of cases with and without reconstruction. We mainly used temporalis muscle flaps and autologous fat to reconstruct the skull base, especially for big tumors with large defects16.There are two views on defect repair. Safaee et al.Postoperative facial palsy is a common complication after surgery. Therefore, the perseveration of the facial nerve is important. Intraoperative electrophysiological monitoring could identify and protect the facial nerve. Although facial palsy was the most common postoperative complication, it was worth that we should perform GTR because facial palsy could recovery to some extent.In conclusion, although PVNS of the TMJ with the extension of the skull base is destructive and leads to intracranial extension, surgery can achieve an excellent outcome, especially for GTR. The most common symptom associated with this condition is pain and swelling. RT may be reserved for extensive or recurrent disease. Because of the relatively high rates of recurrence, a sufficient period for follow-up is necessary.


Malignant peritoneal mesothelioma (MPM) is a sporadic and fatal disease of the peritoneal lining. Its diagnosis has been known to be challenging, time-consuming, and money-consuming. In this paper, we report an MPM case of a 58-year-old man with severe abdominal distension. After he had received all kinds of auxiliary examination, including computed tomography scans of the chest and whole abdomen, examinations of peripheral and pleural fluid, positron emission tomography, and twice fine-needle peritoneal biopsies, his disease still could not be confirmed. Eventually, the patient was diagnosed with MPM through laparoscopic biopsy and IHC. From this case, we concluded that clinicians can gradually discover and diagnose the disease through 1) high platelet and CA125 levels and CT imaging results, 2) cytologic examinations of ascites and pleural fluid, 3) peritoneal biopsies , and 4) histopathological examinations and immunohistochemistry findings. The diagnostic process involving this patient can be an example to demonstrate the effectiveness of various auxiliary examination methods in MPM diagnosis. BAP1) gene, which participates in DNA repair and apoptosis, is related to the occurrence of MPM  is a rare and fatal disease of the peritoneal lining . When mee of MPM , 4. TherOn one hand, due to the low incidence of MPM, there are fewer reports about MPM. Unlike pleural mesothelioma, which accounts for most mesotheliomas, MPM only represents 7%–10% of cases . It has This case report describes the detailed diagnostic process involving a 58-year-old man with significant ascites. We truly experienced the difficulty of MPM diagnosis and would like to demonstrate the effectiveness of various auxiliary examination methods for diagnosing this case.In August 2020, a 58-year-old man was hospitalized in the Center of Gerontology and Geriatrics, West China Hospital, Sichuan University due to chest tightness. The patient’s abdominal CT only showed a small amount of pelvic effusion, and all of the tumor markers were at a normal level Table 1.2; and abdominal circumference, 127 cm. On abdominal examination , we found abdominal distension, negative peritoneal irritation signs, positive mobile dullness, and normal bowel sounds. There was nothing special noted in the lung and heart examinations.Four months later, the patient was readmitted to our department with severe abdominal distension for the past 2 months. The only additional symptom the patient had was poor appetite and reduced food intake. No person in the patient’s family had records of a similar disease. Physical examination yielded the following characteristics: height, 174 cm; weight, 105 kg; body mass index, 34.68 kg/m9/L), total protein (55.1 g/L) and albumin (32.7 g/L) were slightly low, but there was normal liver and kidney function. Tumor markers CA125, CA15-3, and cytokeratin 19 fragment were abnormally high in the blood, pleural fluid, and ascites  demonstrated a large amount of effusion in the abdomen and pelvis, along with a small amount of pleural effusion A–C. Enhbumin 32. g/L wereAfter reviewing the history and results of laboratory and imaging tests, the patient could not be diagnosed with tuberculosis. Thus, we believed there was a high possibility that he had a malignant tumor. Positron emission tomography (PET)-CT was then conducted, and it showed inhomogeneous peritoneal thickening, which was accompanied by abnormally increased glucose metabolism. We were uncertain whether this was a tumor or just inflammation  staining showed PCK (+), CK7 (+), CK5/6 (+), WT-1 (+), HBME-1 (+), and desmin (+), supporting mesothelial cell hyperplasia or mesothelial tumors. However, the reports from the pathological department said that the biopsy tissue was either too small to be fully confirmed or useless. Finally, laparoscopy was performed: there were turbid ascites and extensive adhesions in the abdominal cavity and many nodules (most likely tumor-implanted nodules) on the abdominal wall, omentum, organ surface, and between the organs. The abdominal viscera had adhered to a mass, and the suspected primary tumor could not be detected. The postoperative pathological report for the omentum and abdominal wall indicated malignant mesothelioma. Immunohistochemistry showed tumor cells WT-1 (+), CR (+), CK5/6 (+), HBME-1 individual (+), Des individual (+), CD31 (−), and CDX-2 (−), and the positive Ki-67 rate was approximately 30%, supporting the diagnosis of MPM.As his peritoneal cancer index (PCI) was >30 and he had extensive adhesions in the abdominal cavity, the patient was not immediately recommended to receive treatment with cytoreductive surgery (CRS) + hyperthermic intraperitoneal chemotherapy (HIPEC). Thus, he received four cycles of systemic chemotherapy (SC) with pemetrexed, cisplatin, and bevacizumab. After that, the tumor shrank significantly. He then underwent CRS and continued with two cycles of adjuvant chemotherapy. In more detail, each cycle of chemotherapy was used for 3 days, then 21 days apart; d0 represents the day before chemotherapy; d1, d2, and d3 represent the first day, second day, and third day of SC, respectively. The first and second SC cycles involved pemetrexed 900 mg d1 + cisplatin 50 mg d1–d2, 30 mg d3 + bevacizumab 400 mg d0; the third SC cycle and the twice postoperative SC cycles involved pemetrexed 700 mg d1 + cisplatin 40 mg d1, 30 mg d2–d3 + bevacizumab 400 mg d0 (the doses of pemetrexed and cisplatin decreased due to severe bone marrow suppression); the fourth SC cycle involved pemetrexed 700 mg d1 + cisplatin 40 mg d1, 30 mg d2–d3. Adverse reactions during chemotherapy included bone marrow suppression, immunodeficiency, and dysphagia, which improved after symptomatic treatment. We believed that the patient’s compliance was excellent, as he completed the established treatment plan on time and in quantity. The latest MRI showed that the intra-abdominal tumor had resolved by three-quarters of the original size, and ascites and pleural fluid were absent Figure 2MPM is a hostile disease that has often spread over the entire abdomen and pelvis at diagnosis. In the current case, even though this patient was admitted to the second strongest hospital in China , the finBAP1 expression, which is reported in 55%–67% of instances  is the standard course for MPM, and the epithelioid subtype is the best candidate for CRS plus HIPEC . For inoThere are some limitations to our report. First, we did not indicate the MPM histological pattern of the patient, although it is vital for predicting prognosis. The pathology department provided us with a report that did not specify the histological type; thus, we have not provided the patient’s pathological MPM subtype in this report. Second, the patient underwent many auxiliary examinations to diagnose MPM. It was an unpleasant experience and a considerable expenditure for the man and his family. However, the significance of the case cannot be ignored. First of all, there are few case reports focusing on MPM. The diagnostic process of the case was challenging, and the report is educational. Because we repeatedly obtained uncertain evidence that could not be used to diagnose MPM, the patient went through nearly all the recommended methods. Thus, we believe this case is a valuable example of the effectiveness of various auxiliary examination methods in MPM diagnosis. Lastly, this case indicates that investigations into the treatment modality of MPM are promising for providing better survival. In this case, the patient’s prognosis was surprisingly excellent after receiving SC + CRS + adjuvant chemotherapy treatment.From this case, we learned that MPM can be gradually discovered and diagnosed through 1) laboratory examinations of blood and ascites, such as high platelet and CA125 levels, and imaging tests, such as CT imaging results, to provide information on an abnormal peritoneum; 2) cytologic examination to find unusual cells; 3) peritoneal biopsy, including fine-needle biopsy and laparoscopy biopsy, to retrieve peritoneal tissues; and 4) histopathological examination and IHC findings to confirm the final diagnosis. Moreover, we noticed that the patient achieved a fairly satisfactory survival outcome, even though MPM is a disease with a poor prognosis. Thus, we believe that research focusing on the treatment of MPM holds further promise in the future.The original contributions presented in the study are included in the article/Written informed consent was obtained from the individual(s) for the publication of any potentially identifiable images or data included in this article.XZ contributed to the conception and design, data acquisition and analysis, literature review, article drafting, and manuscript revision. KX did much work acquiring and interpreting data, as she was the patient’s attending physician, and she also took part in manuscript drafting and revision. LZ contributed to the conception and design and literature review and took part in drafting the article. XH guided the whole process, prepared the report, and revised the paper critically and crucially. All authors gave final approval of the version to be published.This case report was funded by the Health and Scientific Research for Cadres in Sichuan Province (Grant no. 2020-103 and 2019-104).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.


Takayasu arteritis is an inflammatory disease of the aorta and its major branches, which results in stenosis and aneurysm formation. Lesions of the abdominal aorta and renal arteries are common. Nevertheless, lesions of the celiac and superior mesenteric arteries are less common. Since the inferior mesenteric artery is usually preserved and functions as a collateral pathway, developing intestinal ischemia is very rare in patients with Takayasu arteritis. In this study, we report the case of a patient with Takayasu arteritis complicated by ischemic colitis. The patient was treated with surgical repair, which resolved the patient’s symptoms. Lesions of the abdominal aorta and renal arteries are also common. However, lesions of the celiac artery (CA) and superior mesenteric artery (SMA) are less common. In the present case, we surgically treated a patient with Takayasu arteritis complicated by ischemic colitis due to the stenosis of the abdominal aorta and CA and occlusion of the SMA.Takayasu arteritis is an inflammatory disease of the aorta and its major branches, which result in stenosis and aneurysm development. Given that the site of inflammation varies from case to case, the clinical manifestations of Takayasu arteritis are diverse. In Japan, the ascending aorta to the arch and its branches are usually affected. Moreover, head and neck symptoms such as dizziness or upper extremity symptoms such as pulselessness are common.The patient was a 43-year-old woman. At the age of 16 years, she was diagnosed with Takayasu arteritis due to fever of unknown origin and carotid wall thickening and had been taking prednisolone for the next several years. Upon resolution of her symptoms, this medication was discontinued. At the age of 40 years, a routine checkup revealed that she had persistently mildly elevated C-reactive protein (CRP) (0.2–0.4 mg/dL) levels and worsening hypertension. Furthermore, a follow-up computed tomography (CT) revealed calcified stenosis of the abdominal aorta and stenosis of the CA and SMA. She was diagnosed with recurrent Takayasu arteritis and administered prednisolone 15 mg/day and methotrexate 12 mg/week. Aspirin 100 mg/day was administered for CA and SMA stenosis. She continued these medications until the age of 43 years when she experienced a sudden onset of left-sided abdominal pain and bloody stool and visited the internal medicine department of another hospital. Blood test results revealed normal CRP and erythrocyte sedimentation rate levels  and a mildly elevated white cell count of 9,980/µL. Colonoscopy showed no rectal lesions. However, longitudinal ulcers were found in the descending colon and diagnosed as ischemic colitis . The retroperitoneal space was approached through a pararectal incision. Descending thoracic aorta–abdominal aortic bypass and CA/SMA reconstruction were performed with partial extracorporeal circulation . On gross examination, both proximal and distal anastomotic sites showed mild atherosclerotic changes, with no obvious wall thickening or adhesion to the surrounding tissue. The anastomotic sites were reinforced with a bovine pericardium patch. The operative time was 357 min, and the intraoperative blood loss was 90 mL. Pathological findings of the aortic wall stained with Elastica van Gieson showed that the elastic tissue was torn off in the fibrotic area. Moreover, there were fibrous growth and mild lymphocyte and plasma cell infiltration in the adventitia and extravascular connective tissue. Postoperative CT showed no anastomotic pseudoaneurysm, and the CA and SMA were patent , followed by the aortic arch (65%) and carotid artery (58%). By contrast, its incidence in the abdomen is slightly lower: 47%–80.2% in the abdominal aorta; 38%–67.5% in the renal artery; and 4.4%–18% and 11%–21.4% in CA and SMA, respectively. Conversely, the IMA is less frequently involved (2%–6.3%). However, symptoms of intestinal ischemia are extremely rare. This rarity is thought to be because the IMA, which can serve as a collateral source, is often preserved even when the CA and SMA are involved. In the present case, although there was stenosis and occlusion in the CA and SMA, no lesion in the IMA was found, which became a well-developed collateral source. Nevertheless, severe stenosis in the abdominal aorta at the level of the diaphragm is thought to have caused ischemic colitis.Besides the systemic symptoms such as fever and malaise (74.9%), head and neck symptoms such as dizziness and headache (47.5%) and upper extremity symptoms (66.4%) are common in patients with Takayasu arteritis. However, other symptoms comprise pain due to intraabdominal aneurysms (including pseudoaneurysms) and pain due to active inflammation of the abdominal aorta, CA, and SMA. Inflammatory bowel disease (IBD) can also cause abdominal pain. IBD in patients with Takayasu arteritis is often difficult to diagnose at the time of initial presentation because endoscopic findings are often nonspecific to IBD. The endoscopic findings in the present case showed longitudinal ulcers in the descending colon, which is a characteristic of ischemic colitis. Moreover, there was no rectal lesion, which is often seen in IBD. The diagnosis of ischemic colitis was established in combination with the contrast-enhanced CT findings.There have been several case reports of patients with Takayasu arteritis accompanied by abdominal pain. However, the causes varied in these cases. Intestinal ischemic symptoms due to abdominal aortic branch involvement were the most frequently reported. In another case, prolonged mesenteric angina resulted in significant weight loss, and the patient was treated intensively. Nevertheless, he did not recover sufficiently and died. Similarly, in the present case, at the first onset of abdominal symptoms, the symptoms were transient and resolved with conservative treatment. Thus, the patient was observed without sufficient evaluation of his abdominal blood flow. At the time of recurrence of abdominal symptoms, contrast-enhanced CT showed worsening of stenosis and occlusion in the abdominal aorta and abdominal branch arteries. Therefore, we decided to perform surgical revascularization. Fortunately, the patient recovered without any sequelae.In patients with Takayasu arteritis, especially when ischemic colitis is present at the time of the first presentation, the rarity of the disease has led to delayed diagnosis and death in some cases. In one case, the rapid progression of intestinal ischemia resulted in extensive intestinal necrosis, and the patient died despite emergency surgery.Ischemic colitis is a rare complication of Takayasu arteritis. If ischemic colitis is the first symptom of Takayasu arteritis, diagnosis and treatment may be delayed, which may result in death due to intestinal necrosis. Furthermore, there are various causes of abdominal pain in patients with Takayasu arteritis. Thus, it is important to perform various diagnostic tests without delay and establish a definitive diagnosis as soon as possible to improve treatment outcomes.


Small bowel obstructions (SBO) are a common surgical problem accounting for up half of all emergency laparotomies in the United States. SBO in the virgin abdomen (SBO-VA) presents surgeons with a unique predicament as historical teaching has mandated operative exploration in these situations due to their association with more sinister etiologies. More recent research has demonstrated that this may not be the case, with adhesive disease comprising the majority of SBO-VA. Small bowel neoplasms however comprise a considerable portion of SBO-VA. Small bowel tumors comprise around 0.5%-2% of all gastrointestinal tumors, with adenomas being the most common type of benign small bowel tumor. These lesions are most commonly encountered in the duodenum, typically involving the peri-ampullary region, Their incidence decreases with descent down the gastrointestinal tract, and are least commonly found in the ileum. Ileal adenomas have been increasingly described in the literature with the rise of advanced imaging and endoscopic capabilities. The vast majority of these lesions remain asymptomatic; however, they have been reported to undergo malignant transformation resulting in obstruction and intussusception. Small bowel obstruction due to ileal adenomas in the absence of malignancy is exceedingly rare, with only one previously reported case in the literature. We describe a case of an SBO-VA secondary to ileal stricture caused by a tubulo-villous adenoma. We then discuss the topics of SBO and SBO-VA, specifically regarding their etiology and historical and modern management, with a particular focus on the diagnosis and management of small bowel neoplasms, specifically small bowel adenomas. Small bowel obstructions (SBO) are a commonly encountered surgical problem resulting in thousands of hospital admissions and resultant operative interventions yearly in the United States. SBOs are most commonly due to adhesive small bowel disease in patients with a previous history of abdominal operations, with up to 70% of these cases resolving with conservative management. Historically, presentation of SBO-VA mandated surgical exploration due to the concern for neoplastic obstruction and the belief that adhesive disease was not present. Recent reviews now demonstrate that adhesive small bowel disease is quite prevalent in SBO-VA. Small bowel neoplasms however comprise a considerable percentage of SBO-VA. We discuss a case of an SBO-VA secondary to an ileal tubulo-villous adenoma that developed a stricture. Following a discussion of the workup and operative management of our case, we discuss an overview of small bowel obstructions, particularly regarding the management of SBO-VA, with an attention towards small bowel adenomas and adenocarcinomas.A 64-year-old male with a past medical history of hypertension presents to our facility with complaints of worsening abdominal pain for the last week. The patient reported that he had been experiencing abdominal discomfort that he initially attributed to gastric reflux, for which he was prescribed anti-reflux medications by his primary care physician, without improvement of symptoms. He stated that the abdominal pain was initially intermittent but subsequently progressed to persistent and constant abdominal pain. He also reported associated abdominal distension during the past week, with nausea and vomiting that began one day prior to presentation. The patient additionally reported not having a bowel movement for the past three weeks. The patient denies previous abdominal operations as well as ever undergoing a colonoscopy or upper endoscopy. He denies any family history of cancer. He reported denied a history of tobacco, alcohol or illicit substance use. He reported his occupation as a mechanic.The patient’s vitals were stable on first encounter, apart from mild tachycardia of 103 beats per minute that normalized with intravenous crystalloid resuscitation. A complete blood count as well as chemistry panel were within normal limits. Lactic acid level was 1.2 mmol/Liter. The patient’s physical exam was significant for abdominal distension that was tympanic to percussion in all four quadrants. The patient’s abdomen was diffusely tender to palpation, with increased tenderness in the epigastrium, but without peritonitis. No hernia was palpated in the bilateral inguinal regions. There were no prior surgical scars on the abdomen or pelvis. Computed tomography (CT) of the abdomen and pelvis with intravenous contrast only demonstrated a high-grade obstruction with dilated, fluid-filled loops of small bowel and a transition point in the right lower quadrant Figure . The traThe patient underwent an exploratory laparotomy where an 8 cm stenotic segment of proximal ileum was noted with decompressed small intestine distally Figure . The steThe patient’s post-operative course was rather unremarkable. The patient was ambulating on post-operative day #1, with bowel function returning on post-operative day #3. The patient was started on a liquid diet, which he tolerated well and was advanced accordingly. The was subsequently discharged on post-operative day #5. Histopathology of the resected segment of bowel demonstrated circumferential tubulo-villous adenoma with ulceration and fibrosis. No foci of carcinoma were noted.Acute SBO is a common surgical problem, accounting for 15% of surgical admissions for abdominal pain in the United States, and accounting for almost half of all emergency laparotomies . ApproxiSmall bowel obstruction in patients with a virgin abdomen (SBO-VA) have historically been operative situations due to the belief that more sinister etiologies are behind the obstruction ,9. CompaSmall bowel tumors are rare, accounting for <3% of all gastrointestinal tumors in the United States . Benign The presence of multiple adenomas is uncommon, but when present typically associated with familial polyposis conditions such as Familial Adenomatous Polyposis (FAP), Turcot Syndrome, Gardner Syndrome, Peutz-Jeghers Syndrome (PJS), and Cronkhite-Canada Syndrome (CCS) -17. MultSmall bowel lesions were historically identified using upper gastrointestinal fluoroscopic studies with small-bowel follow-through (SBFT) -18. SBFTAsymptomatic polyps smaller than 2 cm and those not associated with genetic polyposis syndromes can be observed unless they are amenable to endoscopic intervention -18. AdenOur case report is unique in that there have very rarely been described incidences of ileal adenomas resulting in small bowel obstruction. There have been several cases of ileal adenomas resulting in ileo-cecal or ileo-ileal intussusception ,22. HoweSmall bowel obstructions are an extremely common surgical dilemma. Historically, the management of SBO secondary to adhesive small bowel disease and SBO-VA have been vastly different, with concern for obstructive tumors mandating operative exploration in SBO-VA, especially those without hernia. This paradigm has come into question recently, with recent research demonstrating a relatively comparable percentage of adhesive small bowel disease in both SBO-VA and SBO in the previously operated abdomen. Small bowel neoplasms, however, comprise a considerable percentage of causes of SBO-VA, and should be high on the differential, especially in elderly populations and those with suspicious findings on CT and SBFT. When there is suspicion for small bowel neoplasms, delineation of these lesions can be obtained using advanced imaging modalities including computed tomography enterography (CTE) and magnetic resonance enterography (MRE).Small bowel adenoma and adenocarcinoma are rare gastrointestinal tumors. The majority of small bowel adenomas remain asymptomatic, though these lesions carry a risk for malignant transformation as well as intussusception or obstruction. These lesions are most commonly found in the duodenum, with decrease in incidence with the further distal the small bowel goes. Ileal adenomas and adenocarcinomas are the rarest forms of small bowel adenocarcinoma. Ileal adenomas are increasingly rare and the majority are asymptomatic. When symptomatic, these lesions most commonly present with intussusception due to their close proximity with the ileocecal valve and cecum. Obstructive pathophysiology is extremely rare in ileal adenomas in the absence of adenocarcinoma, with only one other case reported in the literature. When suspected, these lesions should be managed similarly to small bowel adenocarcinoma. Operative intervention is mandated in all symptomatic lesions, as well as asymptomatic lesions >2-3 cm, tumors associated with polyposis syndromes, tumors with features suspicious for adenocarcinoma, and lesions that are amenable to endoscopic intervention.


Fistula is an abnormal tract communicating two epithelial surfaces, uterocutaneous fistula is an extremely rare. The classical presentation is cyclical bleeding from an abnormal opening in the scar of previous cesarean section.A 28 year-old was presented 6 months after cesarean section with history of severe pain and blood discharge during menstruation from the previous transverse supra-pubic scar. The patient had history of previous 2 cesarean sections. Abdominal examination revealed a localized tenderness at site of previous operation scar with 1*1 cm opening at the central part of the scar which was discharging blood during pressure over the lower abdomen. There was no urine discharge from the scar. Vaginal examination by the speculum was normal.Endometriosis was suspected and the patient received medical treatment with little improvement. Later, surgical intervention was done, the scar was excised with an abnormal fistulous tract connecting endometrial cavity at the site of the previous scar was found. Complete excision of the tract was done and the uterus was re-sutured using a slowly-absorbable suture material. The patient had no complications with regular cycles.Uterocutaneous fistula is rare and usually follow cesarean section, suturing the uterus with non-absorbable suture material is reported in this case to be one of the underlying causes, other causes include infection, necrosis, foreign bodies, or malignancy. The fistula tract must be defined and any associated infection controlled, complete resection of the fistulous tract and suturing the uterus with absorbable suture material is required. •Causes of uterocutaneous fistulas are numerous like iatrogenic, endometriosis, malignancy, etc.•The classical presentation is cyclical bleeding from the scar of previous cesarean section.•A combined medical and surgical approach may be required. The exact incidence is not well estimated due to the rarity of this condition Causes of uterocutaneous fistulas are numerous including iatrogenic injuries during surgery, endometriosis, intrauterine devices, chronic infection, malignancies with local invasion to the adjacent organs, prolonged use of abdominal drains, radiation injury, trauma, and the incomplete closure of wounds particularly the uterine wall The classical presentation is that patients present with cyclical bleeding from and abnormal opening in the scar of previous cesarean section Imaging modalities are helpful in the diagnosis which include magnetic resonance imaging (MRI) of the pelvis, hysterosalpingography, fistulography, hysteroscopy, computed tomography scan (CT scan) with intravenous contrast and the use of sagittal reconstruction is another helpful diagnostic tools. Ultrasonography have a limited role in the diagnosis The management depends on the underlying cause and usually directed to individual patients as there is no standard management plan. A combined medical and surgical approach may be required. Hormonal therapy in the form of GnRH (gonadotropin-releasing hormone) agonist will induce atrophic changes in the lining epithelium and may help to reduce the fistula discharge and the subsequent closure of the tract which is helpful in small size fistulas, however this is usually ineffective in large size of fistulas and when the skin opening is large. Surgical intervention is very effective and prompt rapid recovery, laparoscopic surgery may be adopted in selected patients The work has been reported in line with the SCARE 2020 criteria 2A 28 year-old lady who was had previous 3 children  presented 6 months after cesarean section with history of severe pain and blood discharge during menstruation from the previous transverse supra-pubic scar, in between the menstrual cycles she complained from pus discharge from the same site of the scar. The patient had no dysuria with normal color urine, no frequent urination, and the defecation was normal.The patient had history of previous 2 cesarean sections, the past medical history was negative for any chronic illnesses, and the cycles were regular with no history of dysmenorrhea.The cesarean section was done at the 37th week of gestation as an emergency surgery because she had previous 2 scars and developed severe uterine contractions, she delivered a healthy male baby weighing 3.6 Kg.The drug history was negative and the family history was negative for any relevant genetic information or psychosocial abnormalities.2.1On physical examination she had normal vital signs, the patient was afebrile, with no pallor, and the body mass index was 28.Abdominal examination revealed a localized tenderness at site of previous cesarean section scar with a 1*1 cm opening at the central part of the scar which was discharging blood during pressure over the lower abdomen. There was no urine discharge from the scar. Vaginal examination by the speculum was normal.2.2The complete blood count was normal with normal hemoglobin and white blood cells count, the urinalysis was also normal. The patient was sent for abdominal ultrasound which showed no abnormalities.2.3From the clinical scenario we suspected endometriosis as the provisional diagnosis as many patients present after cesarean section complaining from cyclical bleeding at the site of the scar. The patient received medical treatment in the form of GnRH agonists for three months with little improvement and the symptoms recurred after that.The second option was surgical intervention, the operation was performed under general anesthesia, the scar was excised and there was an abnormal fistulous tract which was connected to the uterus and the endometrial cavity at the site of the previous uterine scar which was sutured by a non-absorbable suture material (Silk) at the time of previous surgery , Fig. 2.Complete excision of the tract was done with removal of the previous sutures, removal of the necrotic tissue was done, refreshment of the edges was performed, the uterus was re-sutured using a slowly absorbable suture material, irrigation of the wound was performed with warm normal saline, and then the abdominal wall was closed .Fig. 3AnThe operation was done by a gynecologist and a general surgeon.2.4The immediate post-operative condition was smooth and satisfactory with no immediate complications. Follow up the patient was done for 8 months with no post-operative complications with normal regular menstrual cycles.3Uterocutaneous fistula is rarely seen in clinical practice and usually occur after cesarean sections or other pelvic surgeries. Uterine fistulas occur more commonly with the urinary bladder and the bowel due to iatrogenic injuries, infections, and malignancies. There are some reported cases of uterocutaneous fistulas, the majority of them followed cesarean section Postoperative infection is a major implicating factor for the development of uterocutaneous fistula, sometimes necrosis of the anterior uterine wall may occur due to excessive suturing or infection. Most cases occur in the postpartum period or in the postoperative period, the time of presentation after surgery is not well defined and each patient have a variable presentation. Other causes include endometriosis which cause fistula without previous history of surgery or interventions The diagnosis is usually clinical one when the patient present with cyclical bleeding with menses from the previous lower abdominal wound or chronic purulent discharge of pus or serosanguineous fluid. The injection of methylene blue through the cervix is very helpful when the diagnosis is not very clear, it shows the spillage of the dye from the wound in cases of patent fistulous tract. Imaging can be helpful in detecting the abnormal tract between the uterine cavity and the skin, magnetic resonance imaging (MRI) or CT scan with contrast agents are very helpful to define the anatomical planes in the pelvis. Fistulography or hysterosalpingography are also very informative which involve the injection of the water soluble contrast material through the skin opening or through the cervix and will demonstrate the abnormal connection between the skin and the uterine cavity. Hysteroscopy will visualize the abnormal tract directly Our case presented 6 months after cesarean section due to the use of non-absorbable suture material (Silk) for suturing the uterus, so we can add the use of non-absorbable suture material for closure of the uterus as one of the causes behind the development of uterocutaneous fistula. The use of non-absorbable suture materials increase the risk of chronic infection and may lead to the development of fistula The management of uterocutaneous fistula is a stepwise and involve careful assessment of the patients general condition, investigations to detect the underlying cause, the anatomy of the tract must be identified clearly before any kind of intervention, associated infection must be treated with appropriate antibiotics and drainage of any abscesses, the best surgical management involve excision of the whole fistulous tract with suturing of the uterine wall with an absorbable suture material. Follow up is recommended for early detection of any postoperative event and early intervention when required 4Uterocutaneous fistula is rare and usually follow cesarean section, suturing the uterus with non-absorbable suture material is reported in this case to be one of the underlying causes, other causes include infection, necrosis, foreign bodies, or malignancy. The fistula tract must be defined and any associated infection controlled, complete resection of the fistulous tract and suturing the uterus with absorbable suture material is required.The authors are the main source of funding.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.I was worried about the cause of this cyclical bleeding especially after failed medical treatment.Not applicable.Not applicable.Not commissioned, externally peer-reviewed.Dr. Ayad Ahmad Mohammed is the guarantor of the work.The concept of reporting the case and data recording is done by Dr. Jinan Nori and Dr. Dildar Haji. Drafting the work is done by Dr. Ayad Ahmad Mohammed. Final approval of the work to be published was done by Dr. Ayad Ahmad Mohammed.There is no any conflict of interest.


Teaching Point: In the absence of a clear history of trauma, avulsion of the lesser trochanter should raise a high index of suspicion of an underlying malignancy. Figure 1, arrows). The initial diagnosis of an avulsion fracture of the lesser trochanter was made, in accordance with the professional activities of the patient. Because of aggravating pain and the onset of nightly pain, a follow-up radiograph was performed a few weeks later, showing an expansile radiolucency of the lesser trochanter with an adjacent layered periosteal reaction at the medial cortex of the proximal femoral diaphysis . These findings were suggestive of an aggressive osseous lesion. Repeated MRI showed an osseous lesion causing destruction of the medial cortical bone and a large soft tissue component posteromedial the proximal femoral diaphysis. There was heterogeneous enhancement of the mass . Perilesional edema was present in the iliopsoas and vastus intermedius muscles. The combination of the age of the patient, the absence of a history of trauma, and the aggressive appearance of the osseous lesion with a large soft tissue component was suspicious for Ewing’s sarcoma. After histopathological confirmation, the lesion was treated with neoadjuvant chemotherapy, followed by resection of the proximal femur shaft and placement of a femoral prosthesis. Further follow-up was uneventful.An 18-year-old cyclist was admitted to our hospital for spontaneous onset pain in the left groin for the past months. Computed tomography (CT) showed a fracture at the lesser trochanter of the left femur, which was confirmed on the subsequent magnetic resonance imaging (MRI) (Ewing’s sarcoma is a highly aggressive neoplasm and the second most common bone tumor in children and adolescents with a peak incidence between 10–20 years. The metaphysis of the long bones is most involved (80%), and the femur is the most affected (25%) [Location in the lesser trochanter may rarely mimic an avulsion fracture. The imaging appearance of Ewing’s sarcoma is highly variable and often presents as a large permeative lesion, with lamellated periosteal reaction. Conventional radiography shows an aggressive osteolytic lesion. Computed tomography (CT) is more sensitive for the detection of bony destruction in more complex regions. Magnetic resonance imaging (MRI) is the modality of choice for evaluating local tumor extension and staging. Typically, there is bone marrow replacement with heterogeneous enhancement and a large soft tissue mass. Systemic chemotherapy is the keystone in treatment; additional surgery or radiotherapy can be performed depending on the location and size of the lesion.Underlying malignancy should be excluded in case of avulsion fracture of the lesser trochanter, particularly in the absence of trauma.