Bone fibrous dysplasia is a benign disease of bone tissue dysplasia. Vision impairment is the commonest neurological complication of craniofacial fibrous dysplasia. Most of the vision loss caused by craniofacial fibrous dysplasia is usually a gradual process. Very few present with acute visual impairment as described in our case.We report a patient with fibrous dysplasia presenting rapidly progressive visual loss in the left eye secondary to bone cyst formation. Transnasal endoscopic surgery guided by navigation with drainage and curettage of this bone cyst and orbital decompression resulted in progressive improvement in visual acuity that returned to normal 1 month post-operatively.In cases with acute visual loss due to fibrous dysplasia, emergency surgical treatment should be considered to preserve vision. In the surgical approach, navigation-guided nasal endoscopic surgery may be preferred because of its advantages.The online version contains supplementary material available at 10.1186/s12893-021-01459-x. Bone fibrous dysplasia (FD) is a benign disease of bone tissue dysplasia, which occurs before the age of 30 years old, and the incidence rate of bone fibrous dysplasia in females is higher than that in males . CombineThrough this rare case we’d like to demonstrate that early surgical intervention will lead to a better prognosis in such patients with symptoms of optic nerve compression, while the navigation-guided transnasal endoscopic pathway we use will achieve more accurate location and less tissue damage.A patient was presented at the Outpatient department with complaints of sudden visual loss on the left eye for the duration of 4 days. No other complaints were reported, especially no headaches, orbital pain or diplopia. As for medical histories, the patient complained of having been diagnosed with FD 5 years ago with no history of trauma or other neurological symptoms. The patient’s general health was otherwise excellent.On examination, the patient’s best corrected visual acuity was 6/6 in the right eye and Hand Move/Behind Eye in the left eye, the intraocular pressure is within normal range in both eyes. The patient had no hyperemia in the conjunctiva of both eyes, the corneas were transparent and normal in size and shape. The vitreous body and the lenses were transparent and in-place. The pupil of the right eye was 3 mm in diameter, which of the left eye was approximately 5 mm in diameter. Right-eye indirect light reflex and left-eye direct light reflex were slow, with noted presence of an equivocal left relative afferent pupillary defect (RAPD). The visual field examination showed full field defect in left eye : 33.83 dB P < 0.5%, Pattern Standard Deviation (PSD): 1.89 dB, Visual Field Index (VFI):0%) Fig. , but theA nasal endoscopic optic nerve decompression surgery guided by navigation of left eye was performed after preoperative evaluation with extensive hyperostosis noted in the nasal cavity and sinuses (The video of the operation can be seen in Additional file Postoperative recovery proceeded uneventfully. Physical examination showed her left eye returned to its normal position Fig. . The patThe exact etiology of FD is still unknown . FD is nWhen the optic nerve is oppressed by concurrent cystic degeneration or hemorrhage, which result in vision loss, an operation that decompresses this nerve, can be provided. Diah et al. reported 9 cases of cyst degeneration of craniofacial FD, 4 among them with visual disturbance. Surgery restored vision in two of the four patients, but two others were confirmed to have optic nerve compression and completely lost their vision . Singh eAs for the choice of surgical methods for optic nerve decompression, transcranial and transfacial approaches are classic and universal. Although endoscopic transnasal approaches are not used much, they have existed as early as 1991 . At presAlthough follow-up observation is advocated in the treatment methods for FD, referring to the above data, it can be clearly found that surgical treatment, especially for optic nerve decompression, should be actively performed in acute vision loss caused by craniofacial fibrous dysplasia. Otherwise, the loss of vision will be irreversible. And we should carefully choose the approach of operation according to the patient's condition and wish. In this case, we firstly use navigation system to assist nasal endoscopic decompression surgery, and achieved a good result. The navigation system can help surgeons design surgical paths and accurately locate lesions, together with the surgeon's knowledge and experience, is a good guide to avoid surgical complications.Additional file 1: Video of the surgery guided by navigation.


A 65-year-old woman presented with three days of colicky abdominal pain. Abdominal imaging illustrated small bowel enteritis, ascites in both paracolic gutters, and incidental hepatic steatosis. Although ascites fluid demonstrated high neutrophil count consistent with peritonitis and the patient received adequate antibiotics, she clinically deteriorated. Subsequent exploratory laparotomy revealed necrotic appendix and multiple intra-abdominal abscesses. Histopathology showed acute suppurative appendicitis with multiple other intact small diverticula, indicating likely perforation of inflamed appendiceal diverticula with subsequent abscess formation and abdominal peritonitis. This case highlights the importance of ascites fluid analysis and continued clinical correlation, especially in cases of rare entities with atypical presentations. Diverticula are small protrusions formed from the walls of the tubular gastrointestinal tract and can be associated with several complications such as hemorrhage, inflammation, and perforation. Anatomically, there are two types of diverticula—true and false. True diverticula involve all three layers of the intestinal wall, which consists of the mucosa, submucosa, and muscularis propria, and are most recognized in congenital diverticula such as Meckel's diverticula. False diverticula consist of only mucosa and submucosa without the involvement of the muscularis propria and are associated with acquired diverticulosis. Not surprisingly, many colonic diverticula are false diverticula and originate from weak points in the intestinal wall . Acute aA 65-year-old woman with a history of essential hypertension presented with three days of intermittent, left lower quadrant colicky abdominal pain and nonbilious vomiting, a few hours after an outdoor barbecue, along with subjective fever, chills, and dizziness. Abdominal and pelvic computed tomography (CT) with contrast showed signs of small bowel enteritis . Inciden3 with 75% neutrophils, 12% lymphocytes, 13% mononuclear cell, glucose of 94 mg/dL, protein of 3.9 g/dL, and LDH of 459 IU/L. The patient was continued on cefepime for possible spontaneous bacterial peritonitis (SBP). However, by the third day of admission, the patient's abdominal pain had intensified. The gastroenterology team was consulted for the management of ascites. On examination, the patient had a distended, rigid abdomen with voluntary abdominal guarding and diffuse significant tenderness to palpation suggestive of peritonitis. Given that diagnostic paracentesis was also suspicious for secondary bacterial peritonitis by Runyon's criteria [At admission, the emergency general surgery service was consulted and recommended intravenous hydration and treatment for infectious enteritis under the general medicine service. A diagnostic paracentesis on the second day of hospitalization revealed an ascitic fluid white blood cell (WBC) count of 18,800 cells/mmThe patient was initially treated conservatively for a possible SBO, but due to continued worsening abdominal pain, she subsequently underwent diagnostic laparoscopy. This revealed multiple pockets of pus and purulent peritonitis. Consequently, the laparoscopy was converted to open laparotomy with drainage of intraloop, right lower quadrant, and pelvic abscesses. Appendectomy for a necrotic-appearing appendix was also performed. Histopathology showed acute suppurative appendicitis and periappendicitis with multiple small diverticula along the length of the remaining viable appendix, supporting the likely perforation of one or more diverticula secondary to acute inflammation with subsequent abscess formation and abdominal peritonitis . The patThere are two classifications of diverticula: congenital or acquired. Congenital diverticulum, or true diverticulum, is a rare entity, with minimal incidence of perforation because it includes all the layers of mucosa, submucosa, and muscularis propria. By comparison, an acquired diverticulum is a false diverticulum that does not involve the muscularis propria and therefore has higher tendency for perforation , 7.In retrospect, this patient presented with nonspecific symptoms of appendiceal diverticulitis of false diverticula. The patient's nonspecific gastrointestinal symptoms including abdominal pain and ascites along with initial imaging concerning for parenchymal liver disease led to a suspicion for SBP. Despite antibiotic therapy, the patient continued to worsen with peritoneal signs on abdominal exam and persistent leukocytosis. Moreover, elevated ascitic total protein and LDH were highly concerning for secondary peritonitis by fulfilling two of three Runyon's criteria, which has a sensitivity and specificity of 67 and 96%, respectively . This prYersinia, Campylobacter, and Salmonella and pseudomembranous colitis caused by Clostridium difficile and especially from acute appendicitis, both clinically and radiologically [Delay in the diagnosis and treatment of appendiceal diverticulitis is not uncommon. Appendiceal diverticulitis is often difficult to distinguish from other intra-abdominal infections of the right-sided colon such as infectious ileocecitis caused by ogically . Clinicaogically . A retroogically . Furtherogically . Nonspecogically .Recent studies suggest that there may be subtle characteristics in patient presentation that help differentiate between appendiceal diverticulitis and acute appendicitis. For example, appendiceal diverticulitis more commonly presents in older patients compared to acute appendicitis, with an average of 43 versus 29 years, respectively . These f


CYP11A1 gene encoding the cholesterol side-chain cleavage enzyme (P450scc); the mutations were 940G > A (p.Glu314Lys) and c.1393C > T (p.Arg465Trp). The same mutations were found in his 29-year-old sister, who was then also diagnosed for primary adrenal insufficiency. Deficiency in P450scc is an extremely rare genetic autosomal recessive disorder with around 40 described families in the literature and 30 different mutations. As the diagnosis of delayed onset of P450Scc mutation is difficult, this case illustrates the need for a systematic endocrinological assessment in any case of bilateral testicle lesions, thus avoiding unnecessary surgery.Adrenogenital syndrome is commonly associated with a deficiency in 21-hydroxylase but can be present in other rare enzymatic blocks. We report here the case of a 31-year-old man who presented with bilateral painful testicle lesions leading to bilateral partial orchiectomy as they were suspected for malignancy. These lesions were finally identified as benign testicle adrenal rest tumors (TARTs), and the patient was actually belatedly diagnosed with primary adrenal insufficiency due to 2 mutations of the  Adrenogenital syndrome is commonly associated with an enzymatic block and congenital adrenal hyperplasia (CAH), in which gonadal lesions develop from adrenal-like cells. In these cases, increased levels of ACTH are associated with hyperplasia of ACTH-sensitive tissues, mainly the adrenals, but also within other sites like the testis or ovaries . These tTARTs were mainly described in defects in 21-hydroxylase, which are the most frequent enzymatic blocks representing 90 to 94% of CAH . TARTs cCYP11A1 encoding the cholesterol side-chain cleavage enzyme (P450scc), the first rate-limiting step of steroidogenesis.We report here the cases of a young man and his sister, who were belatedly diagnosed with primary adrenal insufficiency due to mutations of the 2. In his family, his sister's height was 1.58 m, her weight was 52 kg, and her BMI was 20.8 kg/m2. As his mother's height was 155 cm and his father's height was 158 cm, the target height was estimated at 163 cm ((mother + father)/2). The patient was classified as Tanner stage V because the penis was of adult size. Only facial hair was limited requiring only one shave per week. Electrolyte values were as follows: urea 1.9 mmol/L, creatinine 61 µmol/L, sodium 141 mmol/L, and potassium 4.2 mmol/L. Despite the fact that testosterone measurement was not performed, hypogonadism was suggested by bone densitometry, which showed osteopenia; the lumbar T score was -1.8 SD and the femoral T score was -1.2 SD.A 31-year-old man was referred to the urologist for painful bilateral testicular lesions. No other genital lesions were observed, and in particular, no hypospadias. The patient did not report any decrease in libido or erectile dysfunction and had normal ejaculations. The blood pressure was 106/63 mmHg, the heart rate was 67 beats per minute, the height was 174 cm, the weight was 66 kg, and the BMI was 21.8 kg/mUltrasonography revealed bilateral, heterogeneous, and vascularized lesions, localized on the top of both testicles, within the testicle hilus. Testicles were found to be hypotrophic as measured at 6 to 7 ml . The ultrasound report did not point to a benign tumor . The extemporaneous analysis of the tumor suspected a Leydig cell tumor. Therefore, bilateral inguinal testicular exploration with partial orchiectomy was performed in the first instance, according to the Cancerology Committee of the French Association of Urology (CCFAU) recommendations and the European Association of Urology (EAU) recommendations. Bilateral partial orchiectomy was performed to preserve fertility . No preoThe lesions were first considered as benign Leydig cell hyperplasia at anatomopathology. The tumors were measured macroscopically at 2 and 2.5 cm for the right and left lesions, respectively. They were found to be brown to orange and multimicronodular. Microscopically, the tumors were made of eosinophilic cells with regular round nuclei. In some cells, brown pigments of lipofuscin were observed. Calcification and adipose metaplasia were also observed. As the surgeon first considered these lesions as potentially malignant, classical immunohistochemistry was performed in this setting; however, these cells were first difficult to differentiate from normal Leydig cells, as immunochemistry was not specific. Lastly, because of bilateral lesions, testicular adrenal rest tumors (TARTs) were suspected, although no Reinke crystals were seen , 5–7.The patient was then referred to the endocrinologist for testosterone replacement and hormonal evaluation. Presentation was marked by diffuse melanodermia and important asthenia, since adolescence.Patient's hormone profile is presented in CYP11A1 gene analysis by next-generation sequencing (NM_000781 [This hormone profile was not consistent with the diagnostic of the most common enzymatic blocks, as no elevation of any single steroid was observed. However, this was suggesting an enzymatic deficiency at a very early step in steroid biosynthesis. As the patient's adrenals did not appear enlarged on computed tomography, STAR deficiency was highly improbable. The hypothesis of P450Scc deficiency was conducted by TART frequency in enzymatic blocks is estimated between 14 and 86% according to a publication gathering 33 studies in this field, with underestimation probably due to undone screenings . These lThe TART origin could be due to the development of embryonic primordial adrenogenital cells or from urogenital cell ridges . These tA deleterious paracrine effect is also suspected for Leydig cells or neighbour germinal cells, due to a potentially toxic effect of local steroids production . Some blTART lesions are mostly echographycally irregular, hypoechogenic, and homogenous, but can also be heterogeneous, or with a round shape, and rarely iso or hyperechogenic . They caThese lesions are macroscopically firm, yellow to brown-coloured, round, oval or multilobular . It can The patients reported in our paper harbor an extremely rare genetic autosomal recessive disorder with around 40 described families and 30 different mutations –17. TherCYP11A1, suggesting that mutations of the gene coding for P450Scc enzyme should be more frequent than previously thought [As far as our patients' mutations are concerned, Goursaud et al. have reported that the functional consequences of these mutations can explain an uncomplete phenotype . Both mu thought .The diagnosis of the delayed onset of the P450Scc mutation is difficult. Kallali et al. published the case of 3 brothers, among whom one had unilateral orchiectomy at 25 years of age, with the initial conclusion of Leydig cell hyperplasia , 20.This observation illustrates the difficulty to diagnose primary adrenal insufficiency in adults, especially with mild phenotypes. On one hand, when a block is already known in an individual presenting CAH, the diagnosis of TART is commonly and easily made with ultrasonography, with no need for biopsy or partial orchiectomy that should indeed be avoided. On the other hand, the new discovery of bilateral testicular lesions in an adult should evoke a TART and lead to minimal endocrinological exploration. The evolution of unoperated TART is classically a regression under medical glucocorticoid treatment.


We describe the surgical management of a rare pulmonary angiomatoid fibrous histiosarcoma (AFH). A 62-year-old lady presented with shortness of breath and found to have a large left main pulmonary artery defect that was positron emission tomography-avid.Following discussion in a thoracic multidisciplinary team meeting it was deemed unsafe to biopsy considering its intravascular position. The patient proceeded to theatre for a left pneumonectomy. She was successfully discharged home by Day 7. On follow-up the patient is well, and free of malignancy.AFH is an incredibly rare form of sarcoma, and in particular in the thoracic region. We have demonstrated successful oncological resection of a rare intravascular lesion in the pulmonary artery. A 62-year-old lady presented with shortness of breath and found to have a large left main pulmonary artery defect that was positron emission tomography (PET)-avid. AFH is an incredibly rare form of soft tissue tumour of intermediate grade, and accounts for 0.3% of all soft tissue tumours . It was iocytoma , 5. Theriocytoma , 5, 7, 8Following discussion in a thoracic multidisciplinary team meeting it was deemed unsafe to biopsy considering its intravascular position. The patient proceeded to theatre for a left pneumonectomy. She was successfully discharged home by Day 7. On follow-up the patient is well, and free of malignancy. A 62-year-old lady with a past history of sarcoma presented with shortness of breath at rest and minimal exertion. A computed tomography pulmonary angiogram (CTPA) revealed an ovoid filling defect in her left main pulmonary artery, which did not resolve after 3 months of therapeutic anticoagulation on follow-up CTPA. A PET was performed demonstrating an SUV Max of 11.4 indicative of a high-grade malignancy in the left hilum. PET-CT did not demonstrate any other abnormalities to suggest regional nodal disease or wide-spread dissemination. Her case was discussed at a thoracic MDT meeting and a review of images deemed the lesion unsuitable and for biopsy. Due to the highly suspicious nature of the lesion on PET and the patient’s past history of sarcoma, the decision amongst the team was to proceed to a left pneumonectomy.Routine preoperative work up with an anaesthetist revealed normal lung function tests, cardiac function and blood results of kidney and liver function.The patient was brought into the operating theatre and a general anaesthetic was administered. Fibre-optic bronchoscopy was performed demonstrating no compression or invasion. A routine left posterolateral thoracotomy was performed. Inspection of the left thoracic cavity revealed no pleural disease. The hilar tumour was palpable and deemed suitable for resection with reasonable margins. The mediastinal pleura was incised and the pericardium opened, with care to avoid injury to the left phrenic nerve. The inferior pulmonary ligament was then divided in order to release the lung, with only the hilum to divide next. Care was taken with minimal manipulation, so as not to embolize the tumour in the left pulmonary vasculature or across to the right side. The left main pulmonary artery was carefully dissected and then divided proximal to the tumour using vascular staplers. Attention was then directed at the pulmonary veins which were divided using vascular staplers. The left main bronchus was then identified and divided with staplers. Once it was clear that the tumour was well within the margins of the specimen, stations 4 L, 5, 6, 7, 8, 9 and 10 lymph nodes were dissected with care to ensure complete lymph node resection. Traction sutures were placed on the pericardium to prevent it from retracting towards the opposite hemithorax. A 14 French pigtail drain was placed in the left pleural cavity. Routine closure and dressings were administered, and the patient extubated on the operating table and transferred to the intensive care unit.Patient was cleared for the ward and the pleural drain was removed on Day 1, with no significant post-operative issues. She was discharged home on Day 7 with no post-operative complications.The histopathology was reviewed by four senior pathologists whom concluded that the results favoured the diagnosis of intravascular angiomatoid fibrous histiocytoma. The tumour itself was 25 × 23 × 20mm in diameter –3, predoThis case demonstrates a surgical quandary for the management of a large pulmonary vascular lesion. It both demonstrates that these lesions are rarely safe to biopsy preoperatively to determine origin, grade and prognosis. Intravascular lesions have most frequently been diagnosed incidentally following pulmonary vascular endarterectomy . With suOur case raises the questions on how to both attain preoperative diagnosis, and resect a lesion in this position. We have demonstrated that it is safe and successful to do so with curative intent in the form of a pneumonectomy.


Rectal foreign body insertion has had an increasing trend throughout the years, whereas it is rarely reported in pediatrics. The management and treatment of these cases can become challenging, since it also can present with atypical or even no symptoms in physical evaluation. A 14-year-old boy was referred to our hospital with a history of insertion of a paintbrush into his anus four weeks before the admission. The paintbrush had perforated the colon and was in the abdominopelvic cavity; however, no symptoms of peritonitis were observed. Rectal examination, sigmoidoscopy, and colonoscopy were unremarkable. Exploratory laparoscopy was performed, and the paintbrush was taken out completely. The patient was discharged in good condition. In cases with rectal foreign body insertion, perforation without causing peritonitis or acute abdomen is possible. In these conditions, imaging examinations play an essential role in managing the patients, and laparoscopy can be a proper procedure for retrieving the foreign body. Management and treatment of rectal foreign body (FB) insertion are among challenging conditions due to the variety of objects with different shapes and, as a result, different trauma patterns to the rectum and colon . In the A 14-year-old boy had been visited by a doctor due to the rectal insertion of a paintbrush, in which computed tomography (CT) of the abdomen was performed and demonstrated a 90 × 6 mm shaped object in the abdominopelvic cavity. He was referred to our hospital for further evaluation.He reported a history of rectal insertion of a paintbrush four weeks ago, which resulted in rectorrhagia and pain during defecation since that time. The 4-week delay in seeking medical care was because of the patient's mortification. In physical examination, no fever, abdominal tenderness, or rebound was observed. In his CT image, the paintbrush was in the abdominopelvic cavity, located in the middle part of the lower abdomen. Oblique orientation of the FB was seen with the left tip pointing to the left lower abdomen and the right tip to the posterior of the lower abdomen, just anterior to sigmoid bowel loops. The location of the FB was reported in the mesenteric area with adjacent fat stranding and not in the bowel loops . HoweverThe patient was taken to the operating room for general anesthesia and was placed in the supine position. Insufflation was done via the insertion of a Veress needle into the umbilicus. Then, a trocar was passed from the umbilicus for the laparoscope, and two laparoscopic graspers were passed from the abdominal wall, one in the right lower quadrant and the other in the left lower quadrant. The peritoneal cavity was explored, and the tip of the paintbrush was found. The paintbrush was covered with omentum, so dissection of the omentum was performed, and the paintbrush was taken entirely out by laparoscopic graspers . No otheThe patient received ceftriaxone (25 mg/kg/dose every 12 hours for two days) and metronidazole (10 mg/kg/dose every 8 hours for two days) postoperatively and was discharged in good condition three days after the surgery. The patient has been visited routinely at our clinic postoperatively, with no signs and symptoms presented. After three years from the operation, he is well without any complaints.The trend of rectal FB insertion is increasing based on studies from western countries . The majThere were two points in our case, which were interesting to be mentioned: (1) the paintbrush had perforated the colon; however, the site of perforation healed, and there was no evidence of any injury in sigmoidoscopy and colonoscopy. (2) Although the paintbrush entered into the peritoneal cavity, there were no symptoms of acute abdomen or peritonitis.Few studies of pediatric rectal FB insertion are reported in the literature. A detailed history and physical examination are crucial in the first step . PhysicaImaging is essential for retained FBs. Although radiography is suggested to be the first diagnostic approach, CT imaging can give more information, especially in the presence of peritoneal symptoms or in higher FBs. Typically, colorectal FBs with perforation can be diagnosed with wall disruption of the bowel and the FB position in the image; however, free gas is not commonly seen. Besides, colovesical fistula or inflammatory mass can be present .There are various therapeutic approaches, including transanal extraction, endoscopic technique, and surgical procedure, for retained FBs . FB shouThere are few reports regarding the management of pediatric rectal FBs in the literature . We propIn conclusion, our case demonstrated that perforation of the colon and retained FB in the peritoneal cavity may present without acute abdomen or peritoneal symptoms. In these cases, imaging, especially CT scan, can play an essential role in diagnosis and appropriate approach; also, it can be useful for ruling out other conditions. Laparoscopy can be a proper approach in such cases. We suggest a thorough examination before performing any invasive procedure.


Preoperative diagnosis with multimodal approaches might lead to overtreatment. Cautious understanding of cytology and dynamic contrast‐enhanced magnetic resonance imaging is required when a Kuttner tumor is cited as differential diagnosis. Preoperative diagnosis with multimodal approaches might lead to overtreatment. Cautious understanding of cytology and dynamic contrast‐enhanced magnetic resonance imaging is required when a Kuttner tumor is cited as differential diagnosis. Fine‐needle aspiration cytology (FNAC) is still one of the most common methods used for preoperative diagnosis.A Kuttner tumor (KT) is an easily misdiagnosed type of sialadenitis. To alert all head and neck surgeons, we report a case of KT in which an accurate preoperative diagnosis could not be accomplished despite a comprehensive workup.2An 85‐year‐old woman presented with a palpable mass in her right submandibular triangle, which had appeared 3 months prior. She was on medication for hypertension and rarely complained of tenderness. On clinical examination, the mass was stiff and not fixed either to the surrounding skin or to the floor of the mouth. There were no apparent motor or sensory dysfunctions.Dynamic contrast‐enhanced magnetic resonance imaging of the right submandibular gland showed a pattern characteristic of an MT .


Spindle cell hemangioma (SCH), a non-neoplastic reactive vascular lesion, rarely locates in bones. We herein report a successful case of intralesional curettage for an infant with SCH of fibula. An 11-month-old boy was admitted to our center with a painless mass in the right proximal calf. Preoperative digital radiograph demonstrated a massive vascular lesion with an irregular bone destruction of proximal fibula. The lesion was removed via the intralesional curettage approach and pathologically diagnosed as SCH. The patient gained bone structure recovery of right proximal fibula. Two years after the surgery, he experienced no local recurrence. For the management of SCH of fibula with partial bone destruction, we suggest early-stage intralesional curettage as its safety and effectiveness. Spindle cell hemangioma (SCH), characterized by cavernous blood vessels and spindle cell proliferation, has recently been considered as a non-neoplastic reactive vascular lesion , 2. SCH An 11-month-old boy patient presented to our center with a 2-month history of painless mass in the right proximal calf. The mass had been noted to be slowly enlarging in 3 weeks after presentation. No significant symptom was found in this patient. Initial workup performed included radiograph, three-dimensional CT (3d CT) reconstruction, and MRI. Radiographs of the right tibia and fibula indicated an irregular bone destruction of proximal fibula , and theConsidering partial bony structure of proximal fibula was normal, intralesional curettage was performed on the right proximal fibula under general anesthesia. After lesion exposure, a 4.02 × 0.0-cm-sized vascular mass was identified with extension to proximal fibular. Gross examination showed a reddish spongious solid mass, containing topical hemorrhage, partial thrombosis, and irregular bone destruction . With prThis case is rare in comparison with majority of reported SCH cases and merits discussion on following points: location of lesion, selection of surgical intervention, histopathologic characteristics, and long-term postoperative follow-up. SCH is a benign vascular lesion which generally locates in the subcutis at the distal extremities and presents as solitary and multifocal masses. It also can be associated with several clinical syndromes, among which Maffucci syndrome is the most common , 9. In sTo date, the main treatment choice for fibular tumor is segmental or subperiosteal resection, in case of local recurrence at surgical site –19. WideThe histologic appearance in this case consisted of the fissure-like vessel lumens lined with flattened endothelial cells among the spindle cells, which arranged in fascicular pattern in solid area. CD31, CD34, and ERG, as vascular endothelial markers, were reported positive expression in various kinds of vascular tumors. In this case, immunohistochemical analysis revealed positive staining for CD31, CD34, and ERG in the majority of spindle cells, consistent with the diagnosis of SCH , 22. MetIn conclusion, for SCH of fibula with partial bone destruction, intralesional curettage renders a safe and efficient intervention at early stage. Meanwhile, our study was limited by lacking further evaluation for treatment approaches owing to small sample size. Long-time and consistent follow-up could establish the efficacy of our management.The original contributions presented in the study are included in the article/supplementary material, further inquiries can be directed to the corresponding author.The studies involving human participants were reviewed and approved by the Ethics Committee of the Children's Hospital of Nanjing Medical University. Written informed consent to participate in this study was provided by the participants' legal guardian/next of kin. Written informed consent was obtained from the minor(s)' legal guardian/next of kin for the publication of any potentially identifiable images or data included in this article.XZ revised the manuscript and approved the final manuscript as submitted. RW performed the surgery and conducted the data analyses. TH wrote sections of the article and edited the figures. TH and RW contribute to this work equally. All authors read and approved the final manuscript.The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.All claims expressed in this article are solely those of the authors and do not necessarily represent those of their affiliated organizations, or those of the publisher, the editors and the reviewers. Any product that may be evaluated in this article, or claim that may be made by its manufacturer, is not guaranteed or endorsed by the publisher.


A schwannoma is a common benign tumour that can arise anywhere in the body. When it occurs in an unusual location such as the larynx, its differentiation from other tumours can be challenging. Herein, we report a case of a laryngeal schwannoma with extralaryngeal extension that mimicked a thyroid tumour, focusing on its characteristic features on MRI.T2 weighted MRI showed slightly low signal intensity at the central part of the tumour and high signal intensity at the peripheral part of the tumour. Pre-operative imaging suggested that the tumour originated in the thyroid gland. Left thyroidectomy with tumour excision was performed; the tumour was diagnosed as a laryngeal schwannoma with extralaryngeal extension, compressing the thyroid gland. In retrospect, features such as the dumbbell-shape and known as ‘target sign’ on T2 weighted MRI were typical features of schwannoma. Additionally, the tumour’s extension pattern was similar to previous reports of laryngeal schwannomas with extralaryngeal extension.A 19-year-old male presented with a mass in the left side of the neck and hoarseness for 2 years. Endoscopy showed a submucosal mass in the laryngeal region. MRI found a well-defined solid mass in the thyroid gland, extending to the larynx through the lower edge of the thyroid cartilage. A large laryngeal schwannoma may extend outside the larynx with significant compression of the thyroid gland. Understanding the pattern of extension and familiarity with the features on MRI can improve the preoperative diagnosis accuracy. Those that grow in size and extend to the thyroid gland are extremely rare.1,2 Only three previous cases of laryngeal schwannomas with extralaryngeal extension have been reported1,3,4; therefore, little is known about their characteristics on MRI. We report the MRI findings of a laryngeal schwannoma with extralaryngeal extension, focusing on the pattern of extension and the MRI features in an effort to improve preoperative diagnostic accuracy.Schwannomas are common benign tumours derived from Schwann cells in the peripheral nervous system.A 19-year-old male presented with a mass in the left side of the neck and hoarseness for 2 years. There was no family or other medical history relevant to the main complaint in this case. Laryngoscopy showed a submucosal mass in the laryngeal region. The tumour had compressed the left vocal cord.T2 weighted MRI (T2WI) showed a well-defined mass in the left lobe of the thyroid gland extending to the paralaryngeal region through the lower edge of the thyroid cartilage -positron emission tomography (PET) showed mild to moderate FDG uptake in the tumour 3,4 and 1 was a schwannomatosis.1 In the present case, there was no family history suggestive of NF2 or schwannomatosis, but additional MRI showed a vestibular schwannoma located from the internal auditory canal to the cerebellopontine angle. Our case consisted of a pathologically confirmed non-dermal schwannoma and unilateral vestibular schwannoma, suggested schwannomatosis as the clinical diagnosis.6 All cases of laryngeal schwannoma with extrapharyngeal extension, including the present case, were associated with NF2 or schwannomatosis.Laryngeal schwannomas are commonly derived from the internal branch of the superior laryngeal nerve.T2WI corresponds to Antoni A, and the marginal area showing a high signal intensity on T2WI corresponds to Antoni B.5 Based on the MRI findings, the target sign on T2WI was also observed in our case. Compared with CT, MRI can provide a more detailed depiction of internal features, such as the cellularity of the tumour.5 The heterogeneous appearance of schwannomas may be attributed to degeneration and the plethora of cellular components such as cystic and xanthomatous lesions.7 Small schwannomas tend to have homogenous contrast enhancement after gadolinium administration, whereas larger lesions have more heterogeneous enhancement.5 FDG uptake by schwannomas is variable, so it is difficult to differentiate between benign and malignant tumours.8 We emphasize the importance of ascertaining the presence of the tumour extension via imaging. The laryngeal tumour in our case had the typical features of a schwannoma despite its atypical location.Target sign is a well-known characteristic finding of schwannoma on MRI; the central area of schwannoma showing a low signal intensity on 8–10 These dumbbell-shaped schwannomas are characterized by schwannomas arising in a narrow bony foramen and extending outside to form a larger mass than the site of origin. In this case, although the schwannoma did not originate from a narrow bony foramen, it did originate from a narrow space in the larynx and had a dumbbell-shaped morphology.Schwannomas are known to have a dumbbell-shaped morphology, as found in spinal schwannomas and vestibular schwannomas.When a contiguous tumour with no destructive changes in the surrounding area is found both inside and outside the larynx and its characteristics resemble those of schwannomas found elsewhere, the differential diagnosis should include laryngeal schwannoma. Knowing that laryngeal schwannomas can extend from a loose laryngeal structure to the extralaryngeal region is essential. Keeping these features in mind will facilitate accurate preoperative imaging-based diagnosis of laryngeal schwannomas with extralaryngeal extension.A large laryngeal schwannoma may extend to the extralarynx with significant compression of the thyroid gland and may be misdiagnosed as a thyroid tumour. Understanding its pattern of extension and familiarity with its characteristics on MRI will improve pre-operative diagnostic accuracy.A laryngeal schwannoma may extend outside the larynx with significant compression of the thyroid gland without destructive bone changes.T2 weighted MRI shows a characteristic 'target sign', schwannoma should be considered a differential diagnosis even if the mass is in an uncommon location, such as the larynx.When All cases of laryngeal schwannoma with extralaryngeal extension reported so far have been NF2 or schwannomatosis.If NF2 or schwannomatosis is suspected, a previous PET-CT, if available, will provide an opportunity to detect other schwannomas by confirming enlargement of the internal auditory canal or intervertebral foramen with the CT component.


Surgical ciliated cyst is a rare clinicopathological lesion that appears in patients who undergo maxillofacial surgery. In this report we present a particular mandibular case and we discuss the etiopathogenesis and clinicopathological features of this pathology after reviewing the current literature, as well as the origin of its respiratory epithelial profile.The patient is a 67-year-old male with an irregular radiolucency in a previously tooth extracted area of the mandible. The histopathological study revealed a cystic lesion with a connective wall with chronic inflammation, partially lined by a ciliated pseudostratified epithelium. PAS and CK19 stains showed the respiratory characteristics of this epithelium and confirmed the final diagnosis of mandibular surgical ciliated cyst.Surgical ciliated cyst is an uncommon entity associated with maxillofacial surgical procedures with bone and nasal cartilage grafts. In our case, treatment with growth factors present in platelet-rich plasma could explain the respiratory changes observed in the cystic epithelial lining.” The surgical ciliated cyst is an uncommon and benign entity described by Kubo in 1927 that occurs in the maxillary bones that have previously been surgically treated  . This paIt is believed that the origin of the maxillary cases could derive from rests of sinus mucosa that remain trapped in the bone and maintain their proliferative potential . On the In this report, we present the first case of a surgical ciliated cyst of the mandible unrelated to a major maxillofacial surgery and we discuss its main etiopathogenic and clinicopathological aspects, as well the etiology of its respiratory epithelial profile.A 67-year-old man under treatment with antihypertensives and anticoagulants due to cardiac arrhythmia was referred to a dental private practice complaining of a mandibular pain in the fourth quadrant. He had experienced multiple abscesses associated with a fixed dental prosthesis since 2018.Radiographic examination showed the inclusion of 4.5 and a complex endo-periodontal radiolucent lesion on 4.6, with double root fracture and hypercementosis. CT scan revealed an extensive circumferential alveolar bone loss Fig. A. Under The tissue specimen consisted of a dark brown and irregularly-shaped fragment of soft tissue, measuring 1.2 × 1 × 1 cm and brownish cut surface. Microscopic examination showed a thick fibrocollagenous connective tissue wall with different densities and a chronic inflammatory infiltrate Fig. A. The epBased on these data, the final diagnosis of the lesion was surgical ciliated cyst of the mandible. At a one-year of follow-up there was no evidence of recurrence or complications.The prevalence of the surgical ciliated cyst is variable , and it is more frequently located in the upper maxilla , 10. TheAfter performing a literature review on the surgical ciliated cyst of the mandible, we found 12 clinical cases –15  is uncommon , 17. RegWe believe that the platelet-rich plasma (PRP) used for the bone augmentation and the history of a previous chronic severe inflammatory lesion could be the key pathogenic elements of our atypical case. PRP is a concentrated solution containing multiple secreted proteins, such as platelet-derived growth factor (PDGF), transforming growth factor (TGF)-β, endothelial growth factor (VEGF), epidermal growth factor (EGF), fibroblast growth factor (FGF) and insulin-like growth factor (IGF) , 24. It In this special case we think that there are 2 histogenetic possibilities that could explain the appearance of the cyst. The first is that the combination of factors from the PRP and the chronic inflammatory response induced the development of a de novo cyst over the mesenchymal alveolus site after the initial surgery, with extensive areas of respiratory epithelial metaplasia. The second is that remnants of the previous endo-periodontal inflammatory odontogenic lesion remained in the alveolus site after the initial surgery, over which the PRP factors and inflammatory proteins acted, inducing their proliferation, growth and respiratory epithelial metaplasia.In summary, the surgical ciliated cyst of the mandible is an uncommon clinicopathological entity that occurs in patients with history of a maxillofacial surgery, mostly associated with maxillary bone-cartilage grafts. Our case is the first to develop in a patient treated with platelet-rich plasma. Thus, it is very important to make a good clinical history and follow-up of all patients treated surgically in the jaw bones.


Acute cholecystitis and cardiac ischemia can have a similar clinical presentation in some patients. Nonspecific electrocardiographic changes and arrhythmias can occur in acute cholecystitis and gallbladder disease that can confuse the treating physician leading to extensive cardiac workup. Emergency physicians and cardiologists should be aware of these changes so they can expedite the management of acute cholecystitis, which can lead to the resolution of these changes. We report a case of a 53-year-old male who presented with diffuse abdominal pain, nausea, and vomiting. His ECG showed sinus bradycardia. Imaging confirmed the diagnosis of acute calculous cholecystitis. His cardiac workup was unremarkable. His sinus bradycardia was resolved with the management of acute cholecystitis. This case highlights the possibility of a "cardio-biliary reflex" that is initiated by gallbladder pain via autonomic vagal innervation. Acute cholecystitis and cardiac ischemia can have a similar clinical presentation in some patients. Nonspecific electrocardiographic changes and arrhythmias can occur in acute cholecystitis and gallbladder disease that can further confuse the treating physician, leading to extensive cardiac workup. A vagally mediated cardio-biliary reflex is the proposed cause of some of these changes -3. O'ReiA 53-year-old male presented to emergency medical services at the emergency department (ED), complaining of generalized abdominal pain. The pain was colicky and described as mild but continuous. The onset of pain was one day ago. He had nausea with vomiting three times before presentation. He denied any fever, weight loss, anorexia, or change in bowel or urinary habits. There was no significant past medical or surgical history and family history of cardiac disease. He was not using any medications. He denied smoking and alcohol consumption. He had no known allergies. Physical examination showed a temperature of 37°C, blood pressure of 102/64 mmHg, respiratory rate of 18 breaths/min, pulse rate of 33 beats/min, and saturation of 99% on room air. His respiratory and cardiovascular exam was unremarkable. His abdominal examination showed epigastric tenderness with a negative Murphy’s sign. There was no guarding or rigidity. The rest of his physical exam was also unremarkable.An electrocardiogram (ECG) was recorded, which showed sinus bradycardia of 38 beats/min with mild QT interval prolongation Figure . There wReflex sinus bradycardia due to acute cholecystitis or Cope's sign was first described by O'Reilly and Krauthamer in 1971 . Most caIt is postulated that a "cardio-biliary reflex" exists that is triggered by gallbladder pain via autonomic vagal innervation. It can occur in cases of gall bladder disease with or without gall stones .If we consider the patient’s presentation, normal cardiac workup, and resolution of the sinus bradycardia with conservative management of the acute cholecystitis, this patient likely had a case of cardio-biliary reflex. All such cases require special consideration. The altered cardiac rhythm can not only complicate a case of acute cholecystitis but may be part of the initial presentation, thus acting as a confounder to the diagnosis. Further studies may help better delineate this interesting association.


Cervical agenesis or dysgenesis is a rare congenital anomaly. The patients usually present with primary amenorrhoea, primary infertility, and cyclical abdominal pain or with a history of prior surgeries like hymenectomy, vaginoplasty, or adhesiolysis for endometriosis along with well developed secondary sexual characters. We present a case of 15 years old girl with cervical dysgenesis and proximal vaginal agenesis, who presented with severe cyclical abdominal pain. She was managed with cervicovaginal canalization by coring and drilling technique done by vaginal approach with simultaneous laparoscopic guidance. Being a rare type of developmental anomaly of the female genital tract, no standard treatment for type-2 cervical dysgenesis has been established. The patient was followed up for 18 months during which she reported to be having regular menstruation. Cervical agenesis or dysgenesis is a rare congenital anomaly with an incidence of about one in 80,000-100,000 live births ,3.Here we present a case of 15 years old girl with cervical dysgenesis and proximal vaginal agenesis, who presented to our institute with severe abdominal pain. She was managed with cervicovaginal canalization by coring and drilling technique done by vaginal approach with simultaneous laparoscopic guidance. The patient was followed up till eighteen months during which the patient reported to be having regular menstrual cycles.A 15-year-old female was referred to our institute with severe lower abdominal pain. The patient had a history of cyclical abdominal pain and primary amenorrhea. Her height was 145 cm, weight 41 kg and she had normal secondary sexual characters. Examination under local anesthesia revealed a blind-ending vagina of about 5-6 cm in length and the cervix was not visible through the vagina. Investigations revealed normal routine blood tests, hormonal profiles, and karyotype 46XX. Ultrasonography demonstrated a retroverted uterus with hematometra extending up to the upper part of the cervix; non-canalized lower part of the cervix, absence of proximal part of the vagina, and normal adnexa. These findings were corroborated by magnetic resonance imaging (MRI) of the pelvis which demonstrated a normal endometrial cavity with hematometra extending into the upper part of the cervix Figure . The lowThe patient and her attendees were explained about the condition. After proper counseling and written informed consent, the patient was prepared for surgery. Under general anesthesia, the patient was placed in a lithotomy position at an angle of 30° at the hips. Abdominal entry was done with one 10 mm primary port at the umbilicus and four secondary 5 mm ports . On inspection, minimal altered colored blood collection and few endometriotic spots were noted in the pelvis. Bilateral ovaries and other abdominal viscera were normal in appearance. On the posterior aspect, the uterus with cervix was appearing normal in size and shape along with normal ligamentous attachments Figure .The bladder was dissected away from the uterus in the midline Figure . The uppThrough the suprapubic port, a Maryland grasper was inserted into the cervical canal to identify the lowermost end of the cervical canal and the level of obstruction. The bridging tissue between the cervix and blind vagina was delineated and dissected partly by finger dissection through the vaginal route and partly by a Maryland grasper from above Figure , to creaThe Maryland grasper was negotiated through the remaining thin bridging tissue to make a small channel through it. The Maryland grasper was further advanced under finger guidance till it was visible through the vaginal opening. The outer sheath of a 10 mm trocar was loaded over Maryland and advanced gently through the vaginal route to dilate the passage Figure . ConsequA self-fabricated vaginal mold was made over a 5 mL syringe with 14 French Foley catheter at the center and wrapped with sterile gauge pieces and a condom Figure . This moThe post-operative period was uneventful. Vaginal mold was removed on the eighth post-operative day. The patient was discharged on the ninth post-operative day. The catheter was kept in situ for four weeks. Till 18 months of follow-up, the patient reported to be having normal menstruation and on examination, the patency of the newly created track was well maintained.Cervical agenesis or dysgenesis is a rare congenital disorder and limited literature is available about this condition. There is a lack of uniformity in the classification of these kinds of congenital disorders. The complete formation and development of the female reproductive tract have three phases of development; organogenesis, fusion, and septal resorption . Our casIn the case of obstructive uterine anomalies, the normal menstrual flow is occluded resulting in primary amenorrhea as seen in our case. Patients may be asymptomatic or may present with symptoms like hematometra, hematocolpos, and cyclical lower abdominal pain. Sometimes due to retrograde menstrual flow, the patients may present with signs and symptoms of endometriosis, chocolate cyst, and acute emergencies of ovarian torsion .Diagnosis of cervical dysgenesis with proximal vaginal agenesis was made in our case based on history, clinical findings and further confirmed by ultrasonography and MRI. Ultrasonography is usually the first performed diagnostic modality for Mullerian developmental defects, however, MRI is considered as the gold standard . Other cThe treatment and surgical corrective methods are divisive. It depends on the presenting symptoms, type of anomaly, and the level of obstruction. For type-2 cervical dysgenesis, three types of surgical techniques have been described . They arBeing a rare type of developmental anomaly of the female genital tract, no standard treatment for type-2 cervical dysgenesis has been established. Most of the patients present with primary amenorrhea and cyclical abdominal pain. The management of this condition should be individualized keeping in mind the symptoms and the level of obstruction. Proper counseling of the patient and her family about the condition, possible surgical outcomes, and future fertility issues should be discussed before any intervention is planned.


Appendicitis is a common childhood condition requiring surgical intervention and delayed diagnosis can have serious consequences. This report describes the case of a child who presented with an acute abdomen and intestinal obstruction. Multidetector (MD) CT demonstrated a left-sided caecum and an inflamed appendix with a faecolith. Maximum intensity projection (MIP) post-processing was key in identifying the appendicular artery and determine the diagnosis. At surgery, however, a mobile caecum and the appendix were positioned on the right side. The position of the appendix varies considerably depending on the position of the caecum.2 In individuals with situs inversus, midgut malrotation or an unusually long appendix (more than 6.8 cm in children), the appendix may be found on the left side of the abdomen.4 There are a few case reports of CT scan findings demonstrating a perforated appendix in association with caecal redundancy.5Acute appendicitis is a common condition in childhood, but a left-sided appendicitis related to a mobile caecum is rare.A 12-year-old boy presented with generalised abdominal pain, abdominal distension and vomiting for 1 week. There was history of intermittent abdominal pain with constipation, which was managed at a nearby primary healthcare unit. There were no known comorbidities and no history of previous surgery.3 .On clinical examination, the child was ill looking and febrile (38 °C) but fully conscious. The abdomen was distended with generalised tenderness and guarding. Biochemistry was normal except for a raised white cell count of 15 500 mmAbdominal ultrasound demonstrated interloop free fluid and tenderness on probe compression. The appendix could not be identified because of overlying bowel gas in the distended bowel loops. Post-contrast-enhanced abdominal CT scan showed distended small bowel loops with multiple pelvic collections. The caecum was left sided  with a 5Differentials remained as given here. There were no features of midgut malrotation or situs inversus. The referring surgeons were informed and an emergency laparotomy was performed.The surgeons performed a midline incision extending from epigastrium to the suprapubic region. Multiple dilated friable small bowel loops were found. Accidental iatrogenic rupture of small bowel was primarily repaired. The dilated loops were decompressed and the caecum was found on a free mesentery on the right side.The appendix was inflamed with an appendicolith at the base and a ruptured gangrenous tip. Appendectomy was performed and the specimen was sent for histology. Interloop collections were drained followed by peritoneal lavage. There were no adhesions. The patient was discharged after four days later, following an uneventful recovery.Histopathology reported a 7 cm × 3 cm appendix with an occluded lumen and a faecolith.6 The estimated prevalence of a mobile caecum and ascending colon is 10% – 20%.8 During embryological development, a mobile caecum arises from failure of fusion of the colonic mesentery with the posterolateral peritoneum. This results in a free mesentery of the caecum and occasionally the right colon, allowing them to freely move to any part of the abdominal cavity. Occasionally, the caecum may rotate causing volvulus but most of the time it remains in the normal anatomic position.7A mobile caecum is a congenital abnormality with a defective right colonic mesenteric attachment at the lateral peritoneum because of agenesis of the caecal mesocolon.9 Patients with this syndrome may present with chronic intermittent abdominal pain, distension, flatulence, dyspareunia, caecal volvulus and partial or complete bowel obstruction.7Mobile caecum and right colon are commonly present in children as mobile caecal syndrome.10 Meckel’s diverticulum is the most common congenital anomaly of the gastrointestinal tract, present in 2% of the population. It is usually asymptomatic, but 4% – 40% of people with Meckel’s diverticulum may experience complications of diverticulitis, haemorrhage, intussusception, small-bowel obstruction, stone formation or neoplasm. A urachal cyst may become infected and present with abdominal pain, fever, nausea, vomiting, dysuria, voiding difficulty, epididymitis and orchitis.Left-sided acute appendicitis may be difficult to differentiate from an infected duplication, mesenteric or urachal cyst or a Meckel’s diverticulitis.11 Patients with symptomatic mesenteric cysts present with abdominal pain and distension with cysts rarely becoming infected. Duplication cysts of the gastrointestinal system are rare and often associated with vertebral defects, anal atresia, cardiac defects, tracheo-esophageal fistula, renal anomalies and limb abnormalities , especially imperforate anus and hemivertebrae. If symptomatic, they present with abdominal distension, gastrointestinal obstruction or obstipation and caecal duplications, resulting in intussusception.12Mesenteric cysts have an incidence of less than 1:100 000 cases in the general population. They may be located in the mesentery, peritoneum or retroperitoneum. Most are benign but there is a 3% incidence of malignancy.1 Delayed diagnosis may result in complications such as perforation, abscess formation, peritonitis, sepsis, bowel obstruction, infertility and death.13 Chest and abdominal radiographs may be helpful in excluding situs inversus with identification of the heart and gastric bubble in the correct anatomical position. Midgut malrotation may need contrasted upper gastrointestinal studies or cross-sectional imaging. Graded compression ultrasound plays a vital role in the diagnosis of acute appendicitis, especially in children. However, in the presence of bowel obstruction, this modality may be limited by bowel gas. Multidetector CT is the gold standard for imaging acute appendicitis.14 By following the intestinal segments sequentially from the stomach to the anus or vice versa, one is able to demonstrate the location of the caecum and ascending colon.The differential of a left-sided acute appendicitis may not be promptly established in the emergency setting and is often delayed because of atypical clinical signs.7 One limitation of MDCT in acute appendicitis is the overlapping range in maximal appendiceal diameter between inflamed and non-inflamed appendices. The presence of fat stranding, abscesses and an appendicolith are valuable ancillary findings in confirming the diagnosis. In children with minimal abdominal fat, pericaecal fat stranding may not be present, limiting confidence in a definitive diagnosis. The current case demonstrated a left-sided caecum and faecolith associated with an abscess in the left iliac fossa (see Multidetector CT also has the capability to assess the vasculature.ossa see . The preossa see . These v15 Source images are used to display the maximum intensities in the voxels with selection of optimum slab thickness depending on the orientation of the vessels and the density of the adjacent structures. As MIP is not a 3D volume-rendered application, one of the pitfalls is the false interpretation of the relationship of the vessels and adjacent structures in the presence of high-intensity structures.17 This necessitates the user to adjust the display parameters to include the degree of opacification and slab thickness that correctly depicts the vasculature. The MIP requires substantial editing in most cases to clearly depict the anatomical display of the vessels. The quality of the final product may vary according to the experience of the user and the vendor.15Maximum intensity projection software is available on most radiology workstations in South Africa. The MIP provides improved display of vascular maps, improving visualisation of most segments of the vessel including the intraparenchymal branches.This case report highlights MIP as a diagnostic problem-solving tool in the atypical presentation of a mobile caecum. Familiarity with this post-processing software is useful for advanced imaging application in clinical practice. This case report also increases awareness of the mobile caecum as an anatomic variant, which may result in an atypical presentation of acute appendicitis.


Good treatments are available for many cases of vertigo due to a peripheral cause such as benign paroxysmal positional vertigo. Conversely, vertigo secondary to a central lesion remains a treatment challenge typically without good pharmacologic or other treatments. We have successfully treated two patients, the first to our knowledge, with central vertigo, one from brain injury, one after stroke, with low dose olanzapine which we found to quickly and dramatically resolve vertigo and permit functional normalization. In our two cases, we found that a low dose of olanzapine 2.5mg daily  caused vertigo to rapidly and dramatically remit. Interestingly, our two cases had different causes and possibly lesion locations.  Vertigo can have peripheral and central causes . The mosCentral vertigo can occur when there is a lesion or dysfunction of the brainstem vestibular apparatus - vestibular nuclei and their projections, especially to the cerebellum . CentralVertigo secondary to a stroke or brain injury can be symptomatically and functionally disabling and has no consensus treatment . OlanzapCases one and two were presented at the 2018 and 2021 American Academy of Physical Medicine and Rehabilitation Annual Assembly, respectively.Case 1The first case is a 20-year-old male who sustained a gunshot wound to the head. The initial Glasgow Coma Scale was 14, with the unknown duration of loss of consciousness. The bullet entered the left ear, traversed the inferior portion of the left cerebellar hemisphere, and terminated in the midline occipital region. Other significant findings included a subdural hematoma along with the posterior foramen magnum. He underwent a left suboccipital craniotomy for posterior fossa decompression  performed testing, including head thrust test, which was positive for corrective saccades bilaterally, head-shaking nystagmus test, which was positive for dizziness and negative for nystagmus, roll test, which was positive bilaterally left greater than right, and left Dix-Hallpike which was positive for dizziness and vertical nystagmus. This testing revealed motion sensitivity and impaired vestibular-ocular reflex (VOR) consistent with central vestibular hypofunction. From this and the clinical history, it was concluded that the patient's vertigo was of central origin. The patient's symptoms developed into intractable nausea and vomiting, which did not respond to ondansetron, prochlorperazine, scopolamine patch, or meclizine. He could not tolerate therapies or even a clear liquid diet. He had three days of medical misses for therapy due to dizziness, nausea, and vomiting. After more than a week of trialing different medications, olanzapine 2.5mg daily was started, given its off-use label for nausea in cancer patients [Case 2A 53-year-old male presented with severe dizziness, nausea, vomiting, lower limb weakness leading to a fall with a brief loss of consciousness. Initial CT head and cervical spine were negative. Lumbar X-rays showed bilateral minimally displaced sacral wing fractures. Orthopedics recommended weight bearing as tolerated for the right leg and toe-touch weight-bearing for the left leg. The patient's vertigo did not improve with IV hydration and meclizine. MRI of the head revealed an acute infarct in the left medulla and right periventricular white matter Figure  and chroIn acute rehabilitation, his strength was 5/5 on the right side and 4/5 on the left. On initial PT and OT evaluations and for two more days, the patient refused to attempt standing due to vertigo. As in the first case, testing by physical therapy was consistent with central vestibular dysfunction. This, combined with the history and imaging, again led us to conclude that the second patient also had vertigo of central origin. On day 3 of acute inpatient rehabilitation (AIRD3), meclizine 50mg orally twice daily was initiated along with olanzapine 2.5mg oral daily based on its anti-emetic effect [There are good treatments for some peripheral and central causes of vertigo . For exaWe decided to use olanzapine for our patients' central vertigo, given its effectiveness as an anti-emetic . OlanzapLow dose olanzapine was effective in alleviating vertigo in two patients with brain lesions: one from a stroke, the other secondary to brain injury. Future studies of olanzapine for vertigo secondary to stroke or brain injury may be warranted.


Platelet-rich fibrin (PRF) is an autologous blood concentrate obtained without anticoagulants by centrifugation of patients’ peripheral venous blood. PRF is considered to enhance the formation of new bone. The aim of this manuscript was to present two case reports of permanent teeth with closed apexes with periapical lesions, treated endodontically with the use of PRF. The root canals were mechanically cleaned and shaped with NiTi files and irrigated with 5.25% sodium hypochlorite (NaOCl), 40% citric acid (CA), and triple distillated water. Before the canal systems were obturated, A-PRF was used as a scaffold and was placed below the cementodentinal junction with hand pluggers. Cone beam computerized tomography (CBCT) was used to assess the resolutions of periapical radiolucencies. After 6 months, the measurements of both periapical lesions were significantly reduced. Although the performed root canal treatments (RCTs) can definitely be recognized as successful, it must be emphasized that mechanical shaping and cleaning of the root canals with special disinfecting solutions significantly affect the clinical efficacy of RCT. It seems impossible to state that PRF played a leading role in the healing process of the presented periapical lesions. Further studies must be performed to assess whether RCT of mature teeth with an additional PRF application is superior to RCT performed alone. Root canal treatment (RCT) is performed to eliminate pulpal infection, which may be the consequence of severe caries lesions or non-carious conditions, including traumas. The aim of RCT is to remove the inflamed or necrotic pulp, to chemo-mechanically debride the root canal system, and finally to hermetically fill the root canal system with biocompatible material .Four conditions have been found that significantly improve the final outcome of primary RCT, including lack of the periapical radiolucency, homogenous filling of the root canal system, filling of the root canal system that extends to 2 mm within the radiographic apex, and well-sealing post-endodontic restoration . SimilarRCT of teeth diagnosed with endodontic periapical lesions is associated with 49% lower odds of success compared with teeth without periapical lesions . TherefoRegenerative endodontic procedures (REPs) are based on biology and aim to replace impaired dental tissues, including dentin, pulp–dentin complex, and root structures. The major domains that affect regenerative endodontics are growth factors; stem cells; tissue engineering materials; and cell, tissue, and organ culture . SeveralThere are three major types of biological scaffolds used in REPs: blood clot revascularization (BCR), platelet-rich plasma (PRP), and platelet-rich fibrin (PRF) ,7. PRF iAccording to the available literature, PRF seems to be very effective in regenerative dentistry . It is cThe aim of this manuscript was to present two case reports of permanent teeth with closed apexes diagnosed with periapical lesions, treated endodontically with the use of PRF.A 45-year old female patient came to the Department of Endodontic Dentistry Medical University of Gdansk for a severe pain of tooth 23  dental numbering system). Extraoral and intraoral examinations were carried out. The gingiva above tooth 23 was found to be swollen, reddish, and painful to palpation. Tooth 23 showed Grade 3 mobility (Miller’s index of mobility). Examination of the periodontal pocket revealed the presence of exudation, which mostly consisted of pus. The pocket depth (PD) was PD max = 9 mm. Diagnostic examination of a pulp viability was performed with faradic current. Tooth 23 did not respond to electric stimuli, which is typical for nonvital teeth. The periapical tissue condition was additionally tested by the reaction to vertical and horizontal percussion. Both of the reactions were positive.Cone-beam computed tomography (CBCT) was performed. The periapical lesion was measured with the use of CS 3D Imaging v3.5.18 Software . The measured dimensions of the lesion were 9.0 × 7.2 × 9.9 mm. The CBCT images of the periapical lesion are presented in The patient was diagnosed with pulp necrosis with symptomatic apical periodontitis of tooth 23 and qualified for an endodontic treatment. Patient informed consent to per-form root canal treatment was obtained. The endodontic treatment was performed under local anesthesia. The rubber dam was placed before the onset of the endodontic treatment. The root canal was prepared chemo-mechanically with the modified crown-down technique using Nickel Titanium (NiTi) 0.04 rotary instruments  on a working length. The working length (WL) was 27 mm and was confirmed both with the indication of the endometer  and radiologically. Apical gauging was determined with 0.02 hand K-file NiTi ISO 30, so the diameter of apical foramen measured 0.30 mm. The root canal irrigation protocol along with ultrasonic activation (PUI) included the following: 5.25% sodium hypochlorite (NaOCl), 40% citric acid (CA), and triple distilled water. Between the following instruments, the root canal was irrigated with 2 mL of NaOCl with PUI. After the final shape was completed, the root canal was rinsed with 10 mL of NaOCl and 5 mL of CA. The final rinse was carried out with 2 mL of NaOCl and 2 mL of triple distilled water. Every time, the irrigation needle was placed at a depth of 2 mm from the apex.The patient’s blood was drawn from a median cubital vein and collected in glass tubes (each 10 mL). Next, the patient’s blood was centrifugated at 1200 rpm for 8 min in the Neuation iFuge D06 Premium Edition  centrifuge to obtain advanced platelet-rich fibrin (A-PRF). Using sterile tweezers, the fibrin clot was squeezed between two gauze pieces to create an autologous fibrin membrane. After the root canal was dried with paper cones, the freshly prepared A-PRF membrane was placed into the apex and then pushed below the level of the cementodentinal junction using Machtou hand pluggers—size 1/2 NiTi (red) and 3/4 (grey).Finally, the root canal was filled by a thermoplastic method  with calibrated gutta-percha cone MAC ISO 30.04 and AH-plus sealer , using the combination of a Downpack heat source with a Backfill extruder. The methodology of the endodontic treatment was based on the guidelines of the American Association of Endodontists and the European Society of Endodontology ,12. FollThe first follow-up appointment was conducted 1 week after the end of the treatment. The patient reported that the pain after the treatment lasted for 24 h. There were no symptoms of an acute inflammation in the intraoral examination. The gingiva was smooth, pink, and humid, with no pain on palpation. The mobility of the tooth was reduced to Grade 2 Miller mobility index. PD max = 9 mm. The reaction to vertical and horizontal percussion was still positive.The second check-up was carried out 3 months after the end of the treatment. The patient reported that there had been no incidents of pain during that period. No features of inflammation were noticed during the examination. The gingiva was smooth, pink, and humid, with no pain on palpation. The mobility of the tooth was within the physiological limits (Grade 1 Miller mobility index). PD max = 7 mm. The reaction to vertical percussion was positive, but the reaction to horizontal percussion was negative.The third and final appointment was scheduled 6 months after the end of root canal treatment. Intraoral examination revealed healing progression. The gingiva was smooth, pink, and humid, with no pain on palpation. The mobility of the tooth was within the physiological limits (Grade 1 Miller mobility index). The reaction of tooth 23 to vertical and horizontal percussion was negative. PD max = 4 mm. The CBCT images presented healing of the periapical lesion. There was a small area of radiolucency around tooth no. 23 with the dimensions of 2.6 × 1.0 × 0.6 mm. A 42-year old male patient came to the Department of Endodontic Dentistry Medical University of Gdansk because of caries in tooth 23 . The patient was generally healthy. Medical history and extraoral examination were not significant. No soft-tissue abnormality was found in the intraoral examination. Tooth 23 presented Grade 2 Miller mobility index. There was no exudation from the periodontal pocket. The pocket depth was measured; PD max = 3 mm. Tooth no. 23 did not respond to the electric stimuli, which confirmed the tooth was not vital. The reaction to vertical percussion was positive, whereas the reaction to horizontal percussion was negative.The CBCT of tooth 23 was taken. The periapical lesion was also measured with the use of CS 3D Imaging v3.5.18 Software . The measured dimensions of the lesion were 12.7 × 8.7 × 6.4 mm. The CBCT images of the periapical lesion are presented in The patient was diagnosed with pulp necrosis with asymptomatic apical periodontitis of tooth 23. The endodontic treatment was performed with the modified crown-down technique, using NiTi 0.04 rotary instruments  on a working length. The working length (WL) in this case was 29 mm and apical gauging was measured by hand 0.02 K-file ISO 30 and 35, so the diameter of the apical foramen was larger than 0.3 mm, but smaller than 0.35 mm. A-PRF was applied in the same manner as in Patient A. Then, the root canal was filled by a thermoplastic method  with calibrated gutta-percha cone MAC ISO 35.04 and AH-plus sealer . There were also three follow-up appointments, after 1 week, 3 months, and 5 months.One-week follow-up: no pain after the treatment was reported. No abnormalities of the soft tissue were noticed. The mobility of the tooth was within the physiological limits (Grade 1 Miller mobility index). The reaction to vertical percussion was positive.Three months after the treatment: no incidents of pain were observed by the patient. During intraoral examination, the gingiva was smooth, pink, humid, and painless. The mobility of the tooth was within the physiological limits (Grade 1 Miller mobility index). The reaction to vertical percussion was negative.Finally, 5 months after the end of the treatment, there was no evidence of any pathological symptoms. The CBCT images revealed the process of healing of the periapical lesion. There was a small area of radiolucency around tooth no. 23 with the reduced dimensions of 4.6 × 4.6 × 2.1 mm. Within this article, two clinical cases diagnosed with periapical lesions are presented. Both of the cases were treated with conventional RCT (with the principles of the American Association of Endodontists and the European Society of Endodontology) with an additional A-PRF application by the apical foramen to the periapical area. Six months after the end of the performed endodontic treatment, healing of the periapical lesions was noticed. Despite the fact that the performed endodontic treatments can definitely be recognized as successful, it must be emphasized that mechanical shaping and cleaning of the root canals with special disinfecting solutions significantly affect the clinical efficacy of the endodontic treatment. These procedures lead to elimination of the pathogens from the root canals ,14,15. SMoreover, it is known that apical papilla stem cells (SCAPs) are able to survive at apical periodontitis and may even further develop after an endodontic infection ,18. SCAPTherefore, although the presented cases look very promising, it seems impossible to state that PRF played the leading role in the healing process of the presented periapical lesions. Further studies, especially randomized, double-blind controlled trials, must be performed to assess if the endodontic treatment of permanent, mature teeth with periapical lesions with an additional PRF application is superior to the endodontic treatment performed alone.According to the guidelines of the European Society of Endodontology, periapical lesions should be observed for a minimum of four years. If the area of radiolucency remained the same size or the size changes are indiscernible, the endodontic treatment is recognized as a failure and an additional treatment is required, including endodontic surgery, or even tooth extraction . Zhang eAutologous platelet concentrates have been widely used in regenerative endodontics for the treatment of immature teeth. It has been proven that platelet concentrates are able to stimulate apical closure . AlthougSo far, there have not been any manuscripts published analyzing the clinical effects of conventional RCT with supplementary PRF application in the treatment of periapical lesions in mature teeth. Only few case reports have been presented of permanent, mature teeth with periapical lesions treated with RCT combined with surgical procedures, including curettage of the defect and sometimes apical resection ,36,37,38RCT combined with an additional application of A-PRF , performed in permanent, mature teeth diagnosed with periapical lesions, led to a significant decrease in the periapical lesions’ size within six months. However, these observations do not explain the exact role of the A-PRF in the process of healing.


Acute Vasitis a rare condition, but one that can present with a diagnostic dilemma, if not recognized and managed appropriately, may lead to unnecessary surgical interventions with consequent morbidity for the patient. A 27 year old healthy male, presented with left scrotal pain associated with inguinoscrotal swelling.Physical examination revealed left scrotum tenderness and swelling extended along left inguinal area. CT showed multi loculated cystic fluid collection within left seminal vesicle. Acute Vasitis is a rare differential diagnosis for acute scrotum and This case report summarizes the importance of identifying it and how imaging can prevent unnecessary surgical intervention. The pain developed after the patient fell down, then progressively increased in intensity, radiating to the left inguinal area, and associated with inguinoscrotal swelling, nausea and one episode of gross hematuria, patient denied having fever or lower urinary tract symptoms, there was no history of sexual intercourse, previous similar complain and no past surgical history. upon examination the patient was vitally stable, the left scrotum was severely tender and swollen, along with tender mass over the left inguinal area. WBC was 18,000 and other labs were within normal.3Testicular ultrasound showed slightly increased vascularity on the left testis and epididymis with picture of query incarcerated inguinal hernia. therefore, the general surgery tram was consulted. CT abdomen was ordered by general surgery as shown in .Fig. 1(A4Revealed hyperenhancement of the left epididymal head with vascular engorgement, significant thickening, swelling, and fat strandings of the left spermatic cord. This inflammation is seen extending more proximally along the left inguinal canal to involve the vas deference as well as left seminal vesicle which showed increase enhancement compared to the contralateral side. In addition, a small collection was identified in the most inferior aspect of the left seminal vesicle/left ejaculatory duct showed peripheral rim enhancement measuring 1.5*2.8 mm in maximum dimension. Epididymitis, funiculitis and vasitis with small seminal vesical abscess as shown in , Fig. 3 5Vasitis nodsa was first described in 1943 by Benjamin as asymptomatic, chronic inflammation associated with obstruction of vas deference which leads to increase intra-luminal pressure with spermatozoa leakage and inflammatory process.E. coli is thought to be the case of Acute infective Vasitis with negative culture in most of the cases.The ascending spread of urinary pathogens such as H. influenza and Clinical symptoms include asymptomatic nodular lesion in vas deference and are most common in patients following vasectomy.Patients with acute Vasitis may present with localized pain or palpable mass and \ or swelling in the scrotal or inguinal area some of them associated with fever and leukocytosis.It can be easily confused with other deferential diagnosis such as strangulated inguinal hernia, epididymo-orchitis and or even testicular torsion.Usually the diagnosis is unclear, and CT is frequently needed. CT findings include vas deferens thickening, spermatic cord edema and peripheral fat stranding. Literature support the use of CT or MRI to clarify the anatomy and to role out incarcerated inguinal hernia.Acute Vasitis is often diagnosed intra operatively. In our case to the radiologist had a high index of suspicion and requested for a complementary computerized tomography scan to clarify any doubts6Clinical and ultrasound findings of Vasitis, epididymitis, orchitis and inguinal hernia could share some similarities. Further radiologic evaluation by CT Scan is recommended to distinguish between them and avoid unnecessary surgical exploration.None.No conflicts of interest.


Primary pulmonary choriocarcinomas (PPC) are a rare form of extragonadal germ cell tumors (GCT). They present as lung nodules and secrete beta-human chorionic gonadotropin (β-HCG). This is a rare case of PPC that presented insidiously in a postmenopausal woman. Clinical suspicion arose due to markedly elevated serum β-HCG and lung tumor biopsy immunohistochemical staining negative for markers of small cell and non-small cell carcinomas of the lung. The diagnosis of PPC was made after staining positive for markers of GCTs including β-HCG in the absence of a primary tumor in the reproductive organs. The patient was treated with neoadjuvant vincristine, ifosfamide, and cisplatin (VIP) chemotherapy, followed by video-assisted thoracoscopic surgery (VATS) with lobectomy and mediastinal lymph node dissection. This is the first reported case of PPC treated with VIP induction chemotherapy. The patient initially had complete pathologic response and remission; however, she presented with relapse at a nine-month follow-up with new pulmonary nodules and metastatic disease to the brain. Extragonadal germ cell tumors (GCTs) are a heterogeneous group of tumors arising from extragonadal locations without evidence of a gonadal primary. Choriocarcinoma is nonseminomatous and is among the rarest of GCTs. It comprises syncytiotrophoblast cells that secrete β-human chorionic gonadotropin (β-HCG). Although choriocarcinoma is known to metastasize early to the lungs, primary pulmonary choriocarcinoma (PPC) is extremely rare. Only ~65 cases have been reported in the literature, including many diagnosed after autopsy -3. This A 54-year-old postmenopausal female presented to the ED with dyspnea, cough, and intermittent hemoptysis. Physical exam was notable for normal oxygen saturation of 98%, a regular respiratory rate of 16 with unlabored breaths, and clear breath sounds to auscultation. A posteroanterior (PA) and lateral X-ray revealed a 2.5-cm rounded lesion in the left infra-hilar region . A CT chest with IV contrast after completion of the fourth cycle showed a significant decrease in the size of the tumor to 2.1 x 1.6 cm  with lower lobectomy and mediastinal lymph node dissection. Surgical margins and ten lymph node stations were negative for tumor and nodal metastases, respectively. The patient had a complete pathologic response with no residual viable tumor cells. Postoperative recovery was uneventful, and the patient was in remission at four-month postoperative follow-up with undetectable β-HCG.Despite these favorable early results, the patient later presented with relapsing metastatic disease. Approximately nine months after completion of treatment, she presented with new pulmonary nodules and brain metastases. Pathologic examination of the brain lesion was consistent with metastatic choriocarcinoma. Her β-HCG level had also increased to 160 mIU/ml. Despite treatment with adjuvant radiation, her prognosis remains poor.PPC is a rare form of extragonadal malignant GCT that requires the following diagnostic criteria: (1) exclusion of primary lesions in gonads, (2) normalization of β-HCG levels after treatment, and (3) confirmation by pathology . To distDiagnosing PPC is imperative as PPC is known to carry a poor prognosis due to early hematogenous dissemination. The tumor markers α-fetoprotein and β-HCG are essential in managing GCTs. Due to their specificity and sensitivity, they are useful for the initial diagnosis and for the monitoring of treatment along with lactate dehydrogenase -7. Our cThere is no standardized protocol for the treatment of PPC, and there are currently no guidelines as to the proper combination of treatment modalities such as chemotherapy, radiation, and surgery. However, case reports and systematic reviews have focused on achieving remission by combining surgery and chemotherapy regimens adopted from the treatment of high-risk GCTs. Bleomycin, methotrexate and cisplatin (BEP) and etoposide, methotrexate, actinomycin D, cyclophosphamide and vincristine (EMA-CO) have been tried with some success. Our patient did not undergo chemotherapy treatment with EMA-CO or BEP. VIP treatments are another therapeutic option for patients with advanced, gonadal, or extragonadal poor-risk GCTs and are preferred over BEP for mediastinal nonseminomatous GCTs. These patients undergo resection of residual disease with prolonged exposure to oxygen during surgery which can provoke bleomycin-related pneumonitis. Treatment with BEP was considered, but VIP was preferred to avoid this complication that can lead to acute respiratory distress syndrome (ARDS) and higher rates of prolonged postoperative ventilator dependence -12.PPC is a rare lung tumor that should be considered in the appropriate clinical context. Serum β-HCG levels and tissue immunohistochemical stains are essential for diagnosis. To the best of our knowledge, this is the first reported case of PPC that was treated with VIP induction chemotherapy. Longer follow-up and information from other cases will be needed for evaluating outcomes in patients with PPC treated with VIP. Careful consideration of the possible adverse effects of agents such as bleomycin-related pneumonitis is warranted when choosing a chemotherapeutic regimen. Further investigation into proper treatment regimens, as well as factors influencing survival, is needed in order to improve clinical outcomes in patients with PPC.


Crohn’s disease (CD) is a chronic inflammatory disorder which leads to anorectal fistulas. In rare cases, patients develop anal squamous cell carcinoma (ASCC) within these lesions. There is limited literature regarding ASCC in patients with CD. Here, we report on a unique case of advanced verrucous carcinoma (VC), a rare variant of squamous cell carcinoma, developing on the grounds of extensive chronic anorectal fistulas in CD.A 54-year-old male patient with a 20-year history of CD presented with a large inflammatory tumor at the perineal region with multiple discharging perianal fistulas. Histopathological analysis of the perineal mass revealed a VC. Subsequent surgery with radical tumor resection and terminal colostomy resulted in a large perineal cavity and a partially exposed sacrum. The defect extended to a total of 35 × 25 × 25 cm. Reconstruction was achieved through a two-step approach. A first surgical step established an arteriovenous (AV) loop in the upper thigh. Subsequently, a free latissimus dorsi (LD) myocutaneous flap was harvested and anastomosed with the AV loop, allowing for satisfactory closure of the defect and reconstruction of the perianal and perineal region.Radical surgical excision with negative margins is the therapy of choice for VC. This case report demonstrates a curative treatment option with special emphasis on the reconstructive possibilities of a unique case of extended perianal and perineal VC associated with chronic anorectal fistulas in CD. Crohn’s disease (CD) is a chronic inflammatory disorder that may affect the whole gastrointestinal mucosa. Anorectal fistulas are a common manifestation in approximately 23% to 38% of cases of CD and in 60% of patients with CD of the anus or rectum –3. In raThere is limited literature regarding ASCC in patients with CD; most reports consist of small series or single cases , 4–6. Tr2) and a large inflammatory tumor at the perineal region with multiple discharging perianal fistulas  and suffered from alcoholic cardiomyopathy (left ventricular ejection fraction (LVEF) of approximately 20%). Physical examination revealed severe cachexia BMI < 17.0 kg/m7.0 kg/m2Subsequently, abdominoperineal resection, wide excision of the perineal tumor, terminal colostomy, and occlusion of the pelvic floor by omentoplasty and VICRYL® mesh was performed. Surgery resulted in a large wound with a cavity of approximately 35 × 25 × 25 cm and a partially exposed sacrum  loop. For this, the right great saphenous vein was mobilized and anastomosed to the lateral circumflex femoral artery Fig. . SubsequAfter reconstructive surgery, the patient was transferred to the Intensive Care Unit (ICU) and consequently placed in a lateral decubitus position. Mobilization was commenced 7 days after free flap transfer. During the course of the subsequent treatment, the patient developed acute cardiac decompensation (LVEF of < 10%) and consequently acute liver failure, which significantly prolonged the inpatient stay. After stabilization and treatment of cardiac failure, LVEF and liver function gradually recovered. The patient was equipped with a LifeVest® and discharged 69 days after his last surgical procedure  . Adn = 21) . VCs aren = 21) –9. Infecn = 21) , 11. FolRadical surgical excision with negative margins is the therapy of choice for VC , 12, 13.Previous external resection of superior and inferior gluteal vessels as well as the poor quality of local perforator vessels required the installment of an AV loop that could later be mobilized into the defect—thus allowing adequate perfusion for the planned flap –21. Due Reconstructive surgery involving free flap tissue transfer in combination with an AV loop has been reported as single- or two-stage procedure , 20, 22.A free LD myocutaneous flap provides an adequate solution for the reconstruction of large perineal and perianal defects. Moreover, the installment of an AV loop should be considered if locoregional vessels are of inadequate caliber. In the current case, this procedure enabled the curative therapy in a patient with CD, suffering from extensive anorectal fistulas in combination with anal VC.