A radicular cyst is the most common odontogenic cyst of inflammatory origin. Radiographically, it commonly demonstrates clear unilocular radiolucency; radicular cysts with multilocular radiolucency are quite rare.  A 64-year-old Japanese man who presented with a bilocular radiolucent lesion in his left mandible was referred by a dental clinic to our oral and maxillofacial surgery department. He had no particular subjective symptoms. Orthopantomography and computed tomography (CT) revealed an 18 mm × 15 mm lesion with well-defined bilocular radiolucency in the left mandible expanding from the distal side of a canine tooth to the bottom of the 2nd premolar. The lesion included the roots of the 1st and 2nd premolars. The root of the 2nd premolar showed knife-edge resorption. Although the 1st premolar was nonvital, the 2nd premolar was a vital tooth. As differential diagnoses, a radicular cyst, ameloblastoma, odontogenic keratocyst, pseudocyst, and others might be considered. We performed a total resection of the bilocular lesion and diagnosed the lesion as a radicular cyst with tooth structure components inside. The tooth structure components represented lamellar structures of cementum; they were located only in the proximal part (under the 1st premolar) of the lesion. The distal part of the lesion presented distinctive inflammation without tooth structure components.  We encountered a rare case of a bilocular radicular cyst with tooth structure components inside. A radicular cyst is the most common odontogenic cyst of inflammatory origin . It is cA 64-year-old Japanese man who presented with a bilocular radiolucent lesion in his left mandible was referred by a dental clinic to our department of oral and maxillofacial surgery. He had no subjective symptoms. His medical history included coronary vasospastic angina, diabetes mellitus, and hyperlipidemia.Orthopantomography and computed tomography (CT) revealed an 18 mm × 15 mm lesion with well-defined bilocular radiolucency in the left mandible expanding from the distal side of the canine tooth to the bottom of the 2nd premolar. The roots of the 1st and 2nd premolars were included in the lesion. Knife-edge root resorption was observed in the 2nd premolar Figures . AlthougAs a differential diagnosis, radicular cyst, ameloblastoma, odontogenic keratocyst, ameloblastic fibroma, odontogenic fibroma, odontogenic myxoma, jawbone central hemangioma, schwannoma, giant cell granuloma pseudocysts , and hybrid lesions were considered.A biopsy was performed for the lesion under local anesthesia, because the knife-edge root resorption observed in the 2nd premolar suggested the possibility of ameloblastoma. When an ameloblastoma was identified in the biopsy, an adequate margin of safety was necessary for the resection. Based on the biopsy results, the lesion was diagnosed as a radicular cyst with a fibrous cystic wall and nonkeratinized squamous epithelium lining. However, there was a possibility that the incision biopsy did not reflect the entire lesion. We performed a total resection of the bilocular lesion with extraction of the 1st premolar under general anesthesia Figures . The innThe infiltration of inflammatory cells, plasma cells, and lymphocytes was extensive. Epithelial shedding as an effect of the inflammation was also observed Figures . InteresA radicular cyst is the most common odontogenic cyst, accounting for 55% of odontogenic cysts and 52%–68% of all of the cysts of the jaw in humans. Radicular cysts are observed mostly in the third and fourth decades of life, and they have shown male predilection. A “radicular cyst” is defined as a cyst arising from the epithelial residues in the periodontal ligament as a consequence of inflammation following necrosis of the dental pulp. The most common etiology is dental caries with pulp involvement , 2. RadiBefore a pathological diagnosis, in addition to the radicular cyst, a differential diagnosis in similar cases should include ameloblastoma, odontogenic keratocyst, enamel epithelium fibroma, odontogenic fibroma, odontogenic myxoma, jawbone central hemangioma, schwannoma, giant cell granuloma, and pseudocysts  –9. We suIn our patient's case, tumors and pseudocysts were denied as the pathological diagnosis because the lesion had an apparent cystic wall. The whole resected tissue showed a stratified squamous epithelial lining. The wall of the lesion consisted of dense fibrous connective tissue, with an inflammatory infiltrate containing lymphocytes mixed with neutrophils, plasma cells, and histiocytes. These findings are in accordance with the pathology of a radicular cyst . There wAlthough a radicular cyst with tooth structure components has not been reported, it is reasonable to speculate that the cementum-like structure in our patient's case was derived from odontogenic cells present in the wall that, under inflammatory stimulation, started to produce the structure over the years. As another hypothesis, we suspect that the cementum-like structure was derived from tooth fragments by the root canal treatments of the 1st premolar which had been performed several years before. The distal part of the lesion showed severe inflammation without tooth structure components. This suggested that a secondary infection that had occurred in the unilocular lesion caused severe inflammation which led to the formation of a bilocular cyst.In summary, we experienced a rare case of a bilocular radicular cyst with tooth structure components inside. There are numerous reports about radicular cysts, but the multilocular type is quite uncommon. A radicular cyst with tooth structure components has apparently not been reported prior to this case.


Fibroma is a benign tumor. More than 99% fibroma arises from tendon sheaths or tendons. Aggressive Fibroma at posterior compartment knee is a rare case. This report presents one case of fibroma at posterior compartment of the knee with the treatment.A boy with 4-year history of knee discomfort and lump at posterior knee joint, no history of infection and trauma. Physical examination revealed a mass (10 × 9 × 7 cm) with limited Range of Motion extension 30°, flexion 70°. MRI revealed a soft tissue mass at posterior knee compartment. Histologic examination showed a circumscribed and lobulated hypo cellular mass containing spindle cells diagnosed as aggressive fibroma. The patient underwent popliteal resection. An ellipse incision was made 2 cm margin of skin around the biopsy site. Fasciocutaneous flaps were created and retracted to expose the posterior compartment. The tumor had a partial well-defined capsule, popliteal artery and vein embedded and pressing the sciatic nerve. The tumor and enveloping muscles were elevated from the base of the compartment. The sciatic nerve was preserved with epineurotomy. The popliteal artery was reconstructed with saphenous veins graft. After the tumor was resected, the heads of gastrocnemius were sutured to each other and to the hamstring muscles to cover popliteal space. After one year postoperative, there was a good vascularization, no recurrence and neurological deficit with Musculoskeletal Tumour Society Scoring System 80%.One year postoperative with popliteal resection surgery yields a good result. A follow-up is needed for risk of recurrence and malignancy. About 99% of fibromas arise from the tendon sheaths or tendons, and rarely occur in the joint capsules . This fi2A 14-year-old boy presented to our orthopaedic oncology clinic with 4-years history of knee discomfort and lump at posterior knee joint. There was no history of infection and trauma before. A physical examination revealed a mass (sized 10 × 9 × 7 cm) with limited range of motion (ROM) extension 30°, flexion 70° .Fig. 1LuMagnetic resonance imaging (MRI) revealed a soft tissue mass at the posterior compartment of the knee . HistoloThe patient underwent popliteal resection. The ellipse incision was made 2 cm margin of skin around the biopsy site . Fascioc3Fibroma is a rare and benign soft tissue tumor that usually arises in tendons or tendon sheaths . It typiImaging studies with MRI have been reported before. Generally, the T1-weighted MRI typically show a well-defined lesion with homogenous low or iso-signal intensity compared with the muscle. On the T2-weighted images, some cases show a mixture of low and high signal intensity. In our case, the MRI showed a large mass with the size of 10 × 9 × 7 cm in the posterior compartment of the knee.Regarding the histology findings, fibromas infrequently contain hemosiderin-laden macrophages, xanthoma cells or multinucleated giant cells, which in contrast to Giant Cell Tumor of Tendon Sheaths and Pigmented Villonodular Synovitis ,26. NoduRegarding the treatment of fibromas of tendon sheath, the lack of cases made it difficult to build a consensus. The prognosis after marginal excision of these lesions is generally good due to their slow growth and benign histologic appearance. Chung et al. found a local recurrence rate of 24% after excision . However4In summary, fibroma of the knee is a very rare benign soft-tissue tumor. It typically presents as a painless, slow growing, solid nodule. Fibroma should be included in a differential diagnosis of a soft tissue tumor arising from the knee joint. However, the tumor must be excised completely. In our case, one year postoperatively the patient showed a good result but a careful follow-up is needed to observe the risk of recurrence and malignancy.Both authors have nothing to disclosed.Both authors have no source of funding for this article.Ethics approval and consent to publish has been obtained from our institution.Reference number: LB.04.01/A05/EC/035/II/2018.Written informed consent was obtained from the patient’s parents for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.All authors performed the surgery.All authors designed and drafted the manuscript.All authors have read and approved the final version of the manuscriptThis case report is not a “first-in-man” study, thus we did not register the research study.1. Magetsari M.R.N., MD2. Irawan M.N.S, MDBoth authors accept full responsibility for the work and/or the conduct of study.Not commissioned, externally peer-reviewed


Acute appendicitis is known to cause intestinal obstruction. The presentation is commonly due to functional obstruction, but on very rare occasions it presents as mechanical obstruction, especially closed loop.We report a case of a 59-year-old Egyptian man who presented with symptoms suggestive of intestinal obstruction. On examination, he was afebrile with distended tender abdomen with no obvious hernias. There was no history of previous abdominal surgery. Laboratory investigations were within normal range except for elevated serum creatinine levels. Plain erect X-ray and computed tomography scan of his abdomen indicated mechanical small bowel obstruction.in situ. Paralytic ileus was the only postoperative complication which was relieved after 2 days. He was started on orally administered fluids on the third postoperative day, and discharged on the fifth postoperative day.Preoperative preparations with administration of intravenously administered fluids and antibiotics were done for exploratory laparotomy. The operation was approached through a midline incision, revealing dilated small bowel loops with a terminal ileal loop occluded by a ring of his appendix. The appendicular tip was adherent to small bowel mesentery by adhesive band . Release of the band with simple appendectomy was done; a segment of ileal bowel loop was congested but regained its viability after 5 minutes’ application of gauze soaked in warm saline. His abdomen was closed in layers and one drainage tube left Acute appendicitis should be suspected as a cause of mechanical intestinal obstruction in an elderly patient with no obvious diagnostic cause, and can be managed with simple appendectomy when an early intervention is made. Intestinal obstruction is one of the most common surgical emergencies with a very wide spectrum of causes. Acute appendicitis is considered among the causes of intestinal obstruction, particularly functional obstruction. However, few cases of mechanical obstruction following acute appendicitis have been reported, with closed loop obstruction being the rare type. Closed loop obstruction may lead to strangulation and gangrene of the bowel . HoweverWe report a rare case of closed loop small bowel obstruction due to acute appendicitis, managed by simple appendectomy through laparotomy.We review the literature on other cases of intestinal obstruction due to appendicitis with a focus on the closed loop type. We also present a comprehensive discussion about the different presentations, diagnostic approaches, and management.A 59-year-old Egyptian man presented to our Emergency Department with a complaint of absolute constipation for 5 days associated with progressive abdominal distension and pain. Three days later, he could not tolerate any fluids or solid food and began to vomit, with no associated fever, weight loss, or previous attacks of bleeding per rectum. Also, there was no history of any medical illness or previous abdominal surgery.On examination he was alert, afebrile, hemodynamically stable, and with no signs of dehydration. His abdomen was diffusely distended with no scars or any obvious hernias. There was lower abdominal tenderness and hyperperistalsis on auscultation. A digital rectal examination was unremarkable.Laboratory investigations showed elevated serum creatinine level (3.9 mg/dl) whereas white blood cells count, serum sodium level, and serum potassium level were within the normal range.A plain erect abdominal X-ray revealed multiple air fluid levels, which were suggestive of intestinal obstruction. A non-contrast abdominopelvic computed tomography (CT) scan showed small bowel obstruction with a transition zone at terminal ileum while the appendix was not well visualized.He was started on supportive therapy with intravenously administered fluids, antibiotics, and insertion of nasogastric tube and urinary catheter.A decision for exploratory laparotomy was made due to doubtful diagnosis. A midline incision was done under general anesthesia. There were dilated proximal small bowel loops to a point where a long inflamed appendix was wrapped around a loop of terminal ileum. The appendiceal tip was adherent to the ileocolic mesentery, obstructing the ileal loop at two levels causing closed loop obstruction Fig. .Fig. 1InRelease of the adhesive band was achieved by separating the tip of the appendix from the mesentery Fig. . The bowHe suffered from paralytic ileus for 2 days after surgery, which was managed conservatively. He was started on orally administered fluids on the third postoperative day and was discharged on the fifth postoperative day.“Acute appendicitis, with its coincident peritonitis of various degrees of severity, is probably a much more common cause of acute intestinal obstruction than is generally known”; with these words, Lucius Hotchkiss introduced into the medical literature in 1901 three cases of mechanical small bowel obstruction following acute appendicitis. He emphasized mechanical small bowel obstruction rather than the functional type associated with acute appendicitis .Since then, many cases of appendicitis complicated with intestinal obstruction have been recorded. As per the literature reviewed, we found that intestinal obstruction resulting from acute appendicitis is classified pathologically into two subtypes: functional or paralytic subtype and mechanical obstruction subtype.Functional intestinal obstruction results from either: (a) inflammation, the most common type, which is due to the spread of inflammatory exudates and mediators into adjacent structures, such as the ileum and cecum ; or (b) Mechanical bowel obstruction may be classified into either: (a) open loop, which is due to external compression on terminal ileum by inflammatory process in the appendix, either as an abscess or appendicular mass ; or (b) The majority of the reported cases of AT were individuals of 40 years or older including this case study. This can be explained by their diminished immune response to inflammation resulting in different perceptions of pain and abnormal physical findings. The delay in diagnosis for these patients gives more time for repeated attacks of appendicitis with formation of an adhesive band between the appendix and ileocolic mesentery, especially in a lengthy appendix, which eventually obstructs the terminal ileal loop . On the AT occurs more in males than in females; this can be explained by their relatively higher pain threshold that permits repeated attacks of inflammation of the appendix with further wrapping and obstruction of the small bowel , 3.I.Predominant features of appendicitis with some evidence of intestinal obstruction: in this group of patients, intestinal obstruction occurs during the phase of active appendicitis.II.Predominant features of acute intestinal obstruction, while on evaluation/laparotomy found to have appendicitis as the cause. In this group, a history of appendicitis may or may not be apparent.The clinical presentation of this condition can be categorized as Bhandari and Mohandas (2009) reported previously into :I.PredomWe observed that the picture of intestinal obstruction dominates in our case scenario and for a majority of patients with AT as well. Therefore, clinical examination is usually not typical for appendicitis, as only physical findings of intestinal obstruction are evident, while right iliac tenderness is only evident in a few cases and is usually attributed to small bowel ischemia , 9, 10.Laboratory tests performed revealed normal potassium and sodium levels, with elevated level of serum creatinine. A leukocytes count was found to be within the normal range in our study, although it has been reported to be elevated in some series and was mainly attributed to small bowel ischemia , 6, 10. Imaging tests performed in this study were non-contrast abdominal/pelvic CT scan and plain erect abdominal X-ray. The plain erect abdominal X-ray showed air fluid levels with no signs of appendicitis which was comparably the same as in other series. We performed a non-contrast CT scan of our patient’s abdomen and pelvis rather than the contrast type due to high preoperative creatinine levels, to avoid risk of contrast-induced nephropathy.In this study, a CT scan was able to show signs of mechanical obstruction but failed to demonstrate the definite cause of obstruction. Whereas in other series, the use of a contrast-enhanced CT scan was very helpful in demonstrating obstruction, its definite cause, and the presence of bowel ischemia , 7, 9.In most series, patients underwent exploratory laparotomy with diagnostic and/or therapeutic objectives. Inflamed appendix wrapped over the terminal ileum was encountered intraoperatively in almost all cases. The associated ileal loop was gangrenous in a majority of cases due to delayed presentations, necessitating appendectomy and small bowel resection as a definitive treatment , 9.In this case, the bowel was found to be viable intraoperatively, as has been reported in a few series, and this may be due to early presentations of the cases seeking treatments and/or early interventions. Simple appendectomy was found to be sufficient treatment for such cases , 11. LapOne mortality was reported due to postoperative sepsis ; howeverAcute appendicitis may lead to mechanical small bowel obstruction, manifesting as either open or closed loop type (AT). AT occurs mainly in elderly patients with predominant features of intestinal obstruction. Examination and laboratory investigations are not helpful in indicating presence of appendicitis. Imaging may be helpful, especially contrast-enhanced abdominal CT scan.A high index of suspicion for AT is recommended in elderly patients with mechanical intestinal obstruction with no obvious cause. Early surgical intervention prevents the bowel from becoming gangrenous, and appendectomy is sufficient to relieve the obstruction.


Ossification of the yellow ligament (OYL) or ligamentum flavum, usually occurs in the thoracic spine. Focal OYL occurring in the cervical spine is considered rare and is sparsely reported in the literature. We came across a 30-year-old male patient with progressive left upper limb and bilateral lower limb weakness over a period of 3 months, associated with an unsteady gait. Clinical examination revealed bilateral generalized hyper-reflexia in both upper and lower limbs, inverted supinator jerk, Hoffman’s sign and clonus. Myelopathy due to cord compression was suspected and further investigations were done. MRI and CT scans revealed a bony mass in relation to the C6 spinous process projecting anterosuperiorly and narrowing the cervical spinal canal causing cord signal changes from C4 to C6 levels. In view of the deteriorating neurological status, immediate surgery in the form of decompression and posterior stabilization from C4-C6 was performed. Patient gradually recovered after surgery and attained full functional status. We report this case considering the unusual location of OYL and its successful management. The condition was first reported by Polgar in 1920 . OYL in The reason for predisposition of the thoracic spine and frequent association with thoracic prolapsed intervertebral disc herniation in the same spinal segment remains unclear and believed to be of mechanical origin. The condition is slowly progressive and usually requires surgical intervention to prevent subsequent neurological compromise. This article presents a case of cervical myelopathy secondary to OYL in a young African patient. The clinical features, pathogenesis, diagnosis and management are discussed in this report.2.A 30-year old male engineer of African descent, presented with progressive left upper limb and bilateral lower limb weakness over a period of 3 months, associated with an unsteady gait. He complained of altered sensation of his feet, which he described as ‘walking on cotton wool’. He denied any preceding trauma and there was no neck or back pain.Clinical examination revealed that he had normal power in both upper and lower limbs. However, there was generalized hyper-reflexia in all muscle groups of both upper and lower limbs, bilateral inverted supinator jerk, positive Hoffman’s sign and clonus. Sensation was decreased in the left sole of the foot sparing the dorsum. At the time of presentation, patient had a Japanese Orthopaedic Association (JOA) score of 7 and significant gait disability (Grade 3 as per Nurick’s Grading) .Magnetic Resonance Imaging (MRI) of the whole spine showed stenosis with cord signal changes from C4 to C6 levels, secondary to impingement by a prominent bony growth arising antero-superiorly from the C6 spinous process and indenting the spinal canal . ComputeIn view of the above findings, cervical myelopathy resulting from spinal canal stenosis due to OYL was suspected. A posterior decompression laminectomy with excision of the ossified yellow ligament along with posterior instrumentation and fusion from C4-C6 was planned. Lateral mass screws were inserted into C4, C5 and C6. A wide Laminectomy was performed and the OYL was identified. With help of a burr, OYL was freed and removed in a piecemeal manner. OYL was found to be adherent to the dura and care was taken to ensure there was no dural tear and CSF leak , 5.Histological examination of the surgical specimen showed endochondral ossification, lamellar bone and marrow formation within the bony tissue. This was consistent with our diagnosis of OYL. Postoperative period was uneventful, and rehabilitation was initiated as tolerated. Subjective weakness and unsteadiness resolved eventually, and patient recovered to his full functional status by 6 months.3.Compressive myelopathy secondary to ossification of the yellow ligament is an uncommon phenomenon with only a few documented cases in the literature. More than 95% of these cases were reported from Asia, in particular, among the East Asian population. The most common site of occurrence is the thoracic spine leading to thoracic myelopathy –6, 9. A From a surgical perspective, it is important to differentiate ossification from calcification. In OYL, there is formation of mature bone in the ligamentum flavum, characterized by the growth and replacement of the flavum with lamellar bone. In calcification of the yellow ligament (CYL), there is an amorphous calcium crystal deposition within the ligamentum flavum, leading to calcification but no formation of mature bone , 13.In OYL, ossification begins from the capsular part of the flavum, usually forming 2 nodular masses before progressing towards the interlaminar portion at the midline. As the ossification continues, there is unification of adjacent laminae; following which, canal compromise begins from the posterior aspect , 14. ThiDuring the surgery, we performed a wide laminectomy combined with stabilization of the cervical spine with lateral mass screws. A wider surgical field allows for better decompression and visualization of normal anatomy before “peeling off’ the adherent OYL from the underlying dura. Instrumentation and fusion allows for stabilization of the posteriorly decompressed levels and also eradicates the hypermobility which might have been an aetiological factor in the development of the condition –6, 9.4.This case report highlights an uncommon occurrence of a solitary OYL in the cervical spine of a young patient who presented with cervical myelopathy. The combination of CT and MR imaging improves diagnostic accuracy. Wide decompression laminectomy along with stabilization is needed to safely address this pathology. Meticulous care is advised during peeling off the OYL from the adherent dura to prevent possible dural tear and CSF leak.


Introduction: Genital injuries occur in half of cases of sexual assault through digital or penile penetration as well as the use of objects. Women aged >45 years are more likely to have physical injury and anogenital lesions, transmission of STI and HIV. This review focuses on the evidence about surgical reconstruction of the pelvic floor anatomy of adolescents and adult women sexually assaulted during adolescence or adulthood.Method: A systematic literature search was performed in PubMed and Orbis plus for articles published in English and German from June 2008 to June 2018. The literature search was performed in October 2018 by topic combining the following Medical Subject Headings: genital trauma, genital injuries, sexual assault, rape, surgical repair, treatment. Results: 34 records of descriptive studies were identified and 16 full-text articles were included in the present review. Due to the limited number of articles retrieved, articles were not excluded based on methodological design. Superficial genital lesions are common and usually left untreated. For deep vaginal or anal lacerations, intraperitoneal bleeding is usually assessed by means of and additional CT scan or diagnostic colposcopy, cystoscopy, rectoscopy and laparoscopy. Complete reconstruction of the injured is done after. To prevent rectovaginal fistula and uncomplicated primary wound healing a temporary colostomy can be performed.Conclusion: Although most of genital injuries due to sexual assault do not require any major surgical intervention, there is a lack of good quality evidence regarding the best diagnostic and surgical approach to restore deep lesions of genital organs as well lack evidence on contributors to poor wound healing. Therefore, clinical protocols that standardize examination as well as surgical management are encouraged to be developed. Sexual assault and sexual gender violence still occur worldwide, mostly against young women and children. At high risk are also persons who are gay, lesbian, bisexual, or transgendered; physically or mentally disabled; homeless, refugees; alcohol and drug users . Physica It is estimated that one third of survivors never seeks for medical attention and one half only applies 12 hours after the assault . They shPerineal lesions are graded according to the lesion extension in four grades: In the first degree tear, the laceration is limited to the fourchette and superficial perineal skin or vaginal mucosa but the perineal body is intact. In the second degree the laceration extends beyond fourchette, perineal skin, and vaginal mucosa to perineal muscles and fascia; but the anal sphincter and the perineal body are not involved. In the third degree the fourchette, perineal skin, vaginal mucosa, muscles, and internal- or external anal sphincter are torn. In the fourth degree the fourchette, perineal skin, vaginal mucosa, muscles, anal sphincter, and anorectal mucosa are torn . A pre-surgical identification of perineal, anal- and genital lesions is required to determine the medical care that should be provided to the survivor , 2]. It. It2]. IThis review focuses on the evidence concerning surgical reconstruction of the pelvic floor anatomy of women sexually assaulted during adolescence or adulthood. The question addressed was: which surgical approaches are recommended to restore the injured genital organs?A systematic literature search was made in PubMed and Orbis plus for articles published in English and German from June 2008 to June 2018. The literature search was performed in October 2018 by topic combining Medical Subject Headings (MeSH): genital trauma, genital injuries, sexual assault, rape, surgical repair, treatment. Observational or controlled studies were, due to the limited number of articles retrieved, not excluded based on methodological design; consequently case reports were also considered for analysis.46 records of descriptive studies were identified through database searching. To be included, studies needed to provide a description of the surgical approaches used to repair genital trauma of adolescents or adult sexual assault victims Figure 1 . After aAccording to the WHO recommendations , the insThe following order is recommended for genital examination: the mons pubis, inside of the thighs, perineum, anus, labia majora and minora, clitoris, urethra, introitus and hymen. Bruises, scratches, abrasions and tears should be described. If penetration could be occurred, the cervix, the posterior fornix and the vaginal mucosa should be inspected, looking for trauma or bleeding. A bimanual examination and palpation of cervix, uterus and adnexa should be made, looking for signs of abdominal trauma, pregnancy or preexisting infection. If indicated, a rectovaginal examination and inspection of the rectal area for trauma, recto-vaginal tears, preexisting fistulas, bleeding and discharge should be performed. The anal area should be inspected looking for the presence of fecal material on the perianal skin, fissures or rectal tears. Sphincter tone, shape and dilatation of the anus should be evaluated. If there is active bleeding, extended vulvar hematomas, intense pain or suspected presence of a foreign object, supplementary endoscopic examination, colposcopy and vulvo-vaginoscopy should be performed . All feaConservative treatment by means of nonsteroidal anti-inflammatory drugs and cold packs are recommended for superficial, not extensive vulvar hematomas and for perineal wounds , 12]. C. C12]. CIn a prospective study, from 16 children between 4–11 years of age with anogenital lesions after sexual assault, eleven had first and second degree perineal tears like small tears of the hymen, vagina, or the anorectal mucosa, which were managed conservatively . Third aPostoperatively, all cases received thorough on-table irrigation of the distal rectum stump, 7–14 days of broad spectrum intravenous antibiotics and alternate-day under sedation examination with pulse irrigations using an antiseptic solution. Warm genital bath, with antiseptic solution, were provided twice daily up to the seventh postsurgical day. Colostomy could be reverted 4–6 months later. Authors reported that all cases healed satisfactory with exception of two cases, one girl developed a rectovaginal fistula three days after surgery that healed without surgical re-intervention and one case had scarred perineum with unsatisfactory cosmetic outcome. Signs of acute abdomen, hematuria, severe vaginal or rectal bleeding have been described in cases of deep laceration of the third vaginal upper, rupture of vaginal walls or penetrating injuries . In the Rectal injuries above the sphincter should be exanimated under anesthesia to asses the extent of the damage and a diverting colostomy can be needed . One stuGenital and anal injuries requiring surgical care are reported to be low. One study saw 44 females younger than 21 years requiring surgical care for genital or anal trauma out of 11.000 patients which were suspected of having been sexually assaulted. Sexual abuse was reported in 25% of the females, the others were straddle, impalement or motor vehicle accidents . Long term outcomes after anogenital trauma were evaluated by means of a colposcope with 35-mm camera attachment in 94 children, 81 of them girls . It was Partial hymeneal tears, abrasions and contusions healed without or with little signs of previous injury within two to fifteen days, depending on the depth and extension of the lesion . The revAccording to the WHO, most sexual assault victims do not seek acute post-rape medical care. Forensic nurse examiners are often the first clinicians to encounter the survivor . When diInitial visual evaluation is indispensable. If internal injury cannot be ruled out, image- and minimally invasive diagnosis methods such as CT scan, colposcopy, cystoscopy and rectoscopy are reported to be useful to evaluate the damage extension. Diagnostic laparoscopy or laparotomies are usually performed when intraabdominal hemorrhage, organ perforation or any damage proximal of the anal sphincter is identified . Most genital injuries after sexual abuse are reported to be superficial and self limiting, requiring no further evaluation or surgical intervention. Deeper wounds are managed by debridement and primary wound closure to restore structures and organs function . PeritonAdditionally, even the evidence is weak, prophylactic antibiotics are given when puncture, crushing or open wounds were diagnosed , 13]. A. A13]. ASimilar to vaginal cuff dehiscence after hysterectomy , the manThere are few published data that focus on management of genital trauma after sexual abuse. Most reports do not give a detailed description of the surgical approach that is used to repair the injured organs, as well as on contributors to poor wound healing. Therefore, protocols that standardize forensic examination as well as surgical management and psychological support are encouraged to be developed in order to offer a comprehensive care.The authors declare that they have no competing interests.th annual meeting of the German Society for Interdisciplinary Plastic And Reconstructive Surgery 2018, Ulm, Germany.Presentation hold at the 56


The calcifying epithelial odontogenic tumor (CEOT), also known as Pindborg tumor, is a locally invasive benign neoplasm. Histogenesis is controversial.A 26-year-old male presented asymptomatic swelling on the right side of the face, with approximately six months of evolution. At intraoral examination, a hard nodule localized in the right posterior region of the maxilla, measuring approximately 5.0 x 3.0 cm was observed. The computed tomography images showed hypodense lesion with points of calcification associated with two non-erupted teeth. We suspect of odontogenic lesions. The final diagnosis of CEOT was established based on the histopathological aspects.This tumor is more frequent in adult men, usually develops in the posterior region of the mandible and may present clinical-pathological similarities with others odontogenic lesions. The radiological aspects observed in the CEOT are variable and depend on the time of evolution of the tumor. The histopathological examination is mandatory to establish the final diagnosis.Our case was treated with simple enucleation without signs of recurrence in five years of follow-up. Further studies are needed to understand the aetiology and the biological behaviour of this tumor. The oriIn 90% of the cases, the CEOT is intraosseous and extraosseous lesions are common in the anterior gingival region . This tuThe histopathological characteristics are composed of three elements: epithelium, amyloid and calcifications ,6. In adThe aim of this report was to describe the clinical, radiological and histological findings of a case of CEOT that occurred in the right posterior region of the maxilla in a young adult male. The lesion was enucleated and the case has been followed for five years.This case report was written in accordance with the SCARE guidelines .2A 26-year-old man was attended at the Clinical Diagnosis of the Paranaense University (UNIPAR) with the complaint of asymptomatic swelling on the right side of the face, with approximately six months of evolution. Her past medical history was unremarkable.Extraoral examination revealed discrete swelling, hard and fixed in the right side of the posterior region of the maxilla, causing facial asymmetry, with the absence of lymphadenopathy A. IntraoThe radiographic evaluation revealed a radiolucent area related to tooth 17, in addition to expansion and reabsorption of the maxillary vestibular-palatine cortical bone with dislocation of the tooth 18 to the floor of the right orbit. Root resorption of the adjacent teeth was not observed. The coronal computed tomography (CT) shows hypodense lesion extending from the second upper right premolar to the orbit base, with structures compatible with teeth in its interior and some hyperdensing points suggestive of calcification A. In addThe clinical hypotheses of diagnosis were the dentigerous cyst, calcifying odontogenic cyst and CEOT. Fine needle aspiration was negative for fluid. The patient underwent an incisional biopsy. The histopathological analysis of the specimen was compatible with CEOT.The patient underwent enucleation of the lesion under general anesthesia and during the trans-surgical procedure, we detected that the lesion was easily detached from the adjacent bone. The surgical access revealed rupture of the vestibular cortical bone A. On groMicroscopically, the tumor was composed of islands and sheets of polyhedral cells forming intercellular bridges, areas of extracellular and amyloid-like eosinophilic material, small irregular foci of concentric calcifications (Liesegang rings) and cells with nuclear pleomorphism C–F. BaseThe patient has five years follow-up and there were no signs of recurrence.3In the 4th edition of the World Health Organization Classification of Head and Neck Tumors, 23 odontogenic tumors have been listed and CEOT remains among benign epithelial OT subtypes .The odontogenic tumors represent between 2.3% and 4.7% of all lesions diagnosed in specific anatomic pathology services of the buccomaxillofacial complex ,11. The According to the literature ,4 and asThe radiological aspects observed in CEOT are variable and depend on the time of evolution of the lesions, being able to present from a unilocular radiolucent lesion to an entirely radiopaque mass . In our Microscopically, CEOT shows typical characteristics as polyhedral epithelial cells with intercellular bridges, arranged in islands, sheets or cords, with eosinophilic cytoplasm and varying degrees of nuclear pleomorphism, interposed by areas of amyloid material and calcifications (Liesegang rings). In addition, mitotic figures are unusual. It is possible to find variations in the distribution and proportion of the elements that compose this tumor ,5. FurthIn general, the treatment for CEOT is surgical enucleation, but the treatment plan may vary among patients ,7. It haIn summary, we present a case of CEOT in a young adult patient with clinical-radiological characteristics similar to other odontogenic lesions, and histopathological examination was essential to establish the final diagnosis. Our case was treated with simple enucleation without safety margins due to the location of the tumor, trans-surgical aspects and age of the patient, and showed no signs of recurrence in five years of follow-up. In addition, case series studies are needed to better understand the origin and variations in the biological behavior of this tumor.The authors declare that they have no conflicts of interest.There are no sponsors involved in the study.Ethical approval was not required and patient identifying knowledge was not presented in this report. Ethical approval is waived.The written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review on request.All authors contributed signiﬁcantly and in agreement with the content of the article. K. Gruber, L. C. Dogenski and A. C. da Silva Bocassanta participated conducting the patient. S.A.J. de Freitas Filho, L. R. Paranhos and J. P. de Carli participated in the process of writing of the paper. All authors have read and approved the final version of the manuscript.Not applicable.Dr. Luiz Renato Paranhos.Not commissioned, externally peer-reviewed.


A left orbital arteriovenous fistula (AVF) is diagnosed in a patient presenting with proptosis. Intra-orbital AVFs are rare according to the literature search, and therefore, the best treatment modality remains controversial. We present a case of a patient who presented with non-specific symptoms. He was diagnosed with intra-orbital AVF and underwent a trans-arterial embolization. The procedure was complicated by the central retinal artery occlusion, which is one of the most feared complications associated with this procedure. We discuss the modalities in the diagnosis of intra-orbital AVFs as well as stress the importance of an interdisciplinary approach for its timely and efficient management. Intra-orbital arteriovenous fistulas (AVF) are low flow fistulas characterized by direct communication between an artery and a vein bypassing the capillary bed. They are rarely life-threatening; however, in some cases, they may present with a progression of symptoms and may require intervention. Purely intra-orbital AVFs are very rare; diagnosis may be easily missed, and treatment, when symptomatic, remains controversial.We present a case of a patient presenting with non-specific symptoms, in which the rarity of the disease caused a challenge with diagnosis and treatment.A Hispanic male, in his sixth decade of life, with a medical history of hyperlipidemia and hypertension, presented to an ophthalmologist office with complaints of left eye swelling, redness, and bulging. The symptoms were preceded by sneezing and a runny nose three weeks prior. He denied ocular pain or changes in his vision at the time. His ocular pressure was elevated at 24 mmHg and a presumed diagnosis of glaucoma was made. He was referred to the emergency department (ED) for imaging and further management. In the ED, his physical examination was remarkable for left-sided proptosis and conjunctival injection. There was no restricted eye movement or elicited ocular pain. The rest of the physical examination was unremarkable, and the labs ordered, including complete blood count (CBC) and inflammatory markers (C-reactive protein and erythrocyte sedimentation rate) were unremarkable.Imaging performed in the ED included computed tomography (CT) of the brain which was concerning for sinusitis as well as left ocular proptosis. A brain magnetic resonance imaging (MRI) was done for further evaluation which showed enhancement along the supra-ophthalmic vein reflecting possible thrombosis or pseudotumor. However, follow up imaging with magnetic resonance venography (MRV) identified an indirect left cavernous carotid fistula with no evidence of dural venous sinus or left ophthalmic vein thrombosis.The neurosurgery team was involved and a cerebral angiogram was done. The angiogram was remarkable for a left orbital AVF with a connection between the branches of the left ophthalmic artery and the left ophthalmic vein Figures -3. An atThe patient continued to have symptoms of left eye proptosis for weeks after the initial evaluation and eventually developed ocular discomfort. The decision to proceed with the intervention was made on a follow-up visit. He underwent trans-arterial embolization of the AVF. The embolization of the fistula was successful; however, the post-operative course was complicated by the loss of vision in the left eye upon awakening from the anesthesia. A fundoscopic examination was consistent with central retinal artery occlusion which led to subsequent permanent loss of vision of the left eye. AVFs in the orbit are rare. They develop from a single communication between an artery and a vein without a nidus. Most cases are secondary to traumatic events, such as a fracture of the ethmoid bone, or spontaneous. The vast majority of cases have been described in cases after trauma; however, a few cases have been documented spontaneously in patients with a prior history of hypertension, atherosclerosis, and other vascular diseases . Purely Intra-orbital AVFs may be easily miss-diagnosed due to its rarity and common presentation. Symptoms usually involve proptosis, diplopia, ocular hypertension, and engorged episcleral vessels . Vision Carotid-cavernous fistulas are usually high-flow fistulas showing rapidly progressive symptoms. Such fistulas typically require treatment with endovascular embolization or surgical excision. Intra-orbital AVFs are typically low flow fistulas and progression of symptoms is not as dramatic. Conservative management usually includes close monitoring as well as glaucoma medications to lower the intra-ocular pressure . Interventional methods have traditionally involved surgical excision of the fistula or trans-arterial embolization of the fistula. Trans-venous embolization is a newer modality that promises more success rates with less complications. A literature review performed by Lv et al. looked at 26 cases of documented intra-orbital AVFs in the literature who were treated using different modalities. Conservative management was used in nine patients, out of which three reported remission while six reported failure or visual deterioration. Five patients were treated using the surgical approach, out of which two reported cure and three reported failure or visual deterioration. Eleven patients were treated using the trans-arterial approach, out which one patient-reported cure and 10 reported failure or visual deterioration. The trans-venous approach was by far the most successful, with nine out of 13 patients reporting cure [The first case of trans-venous embolization was reported in the year 2000, however, the technique was not successful until the year 2005. Since then, advances in technology and technique have progressed and it seems to carry the highest success rate. Currently, surgical excision is only performed in cases where trans-venous or trans-arterial embolization fails. The trans-arterial approach is still preferred by some physicians based on expertise. The trans-arterial approach seems to carry the highest risk of adverse events. One of the most common complications of this method is occlusion of the retinal artery as was the case for the patient presented in this case report. This occurs due to the close proximity of the arterial feeder to the central retinal artery and the small caliber of the draining branches of the superior ophthalmic vein [When deciding between the trans-arterial and trans-venous approach, it is important to differentiate between an AVF and an AVM. In cases of AVM, the trans-venous approach may result in intra-orbital hemorrhage and worsening of visual function. Cerebral angiography is necessary prior to intervention to fully differentiate between the two entities. The anatomy is also very important. The trans-venous approach is much more cumbersome due to the small caliber and complex, torturous, intra-orbital venous anatomy. In comparison, the trans-arterial approach is much more direct .Our case report presents a rare disease which was a challenge to treat due to its rarity. Multiple treatment options exist; however, they all carry complications and are not performed very frequently. The patient in this case report could not undergo the trans-venous approach due to the small caliber and complex anatomy of his intra-orbital venous system. The trans-arterial approach was performed instead. Unfortunately, the post-operative course was complicated by central retinal artery occlusion which is one of the most common and feared complications associated with this approach. Our case demonstrates the importance of an interdisciplinary approach to the management of intra-orbital AVFs.


Fibroadenoma is the most prevalent benign breast lesion that generally affects middle-aged women; it is rare in adolescents and younger children. The transformation into malignancy is not common. However, multiple recurrences of rapidly enlarging fibroadenomas suggest a high possibility of transforming into phyllodes tumors, which are uncommon fibroepithelial lesions that account for 0.3–0.5% of female breast tumors and typically present in premenopausal women.We report a case of a 17-year-old Syrian girl who previously had three episodes of recurrence of multiple rapidly enlarging fibroadenomas in her left breast and underwent three operations for complete resection of the lesions. However, a few months later, she was readmitted with multiple large masses in the same breast, and pathological findings confirmed a surprising combination of multiple fibroadenomas for the fourth time with a malignant phyllodes tumor (cystosarcoma phyllodes). The patient underwent lumpectomies followed by adjuvant radiotherapy. Long-term follow-up was recommended.Our patient had an extraordinary number of episodes of recurrence at a young age and a rare combination of malignant and benign lesions in the same breast with multiple recurrences. We present her unique, very challenging case with the aim of highlighting the importance of clinical correlation, detailed diagnosis, and careful follow-up. Fibroadenoma (FA) is the most common type of benign breast lesions. It generally affects middle-aged and premenopausal women, whereas it is rare in adolescents and younger children. This tumor is composed of ductal components surrounded by loose fibrous tissue . Giant FA 17-year-old Syrian girl presented to our hospital in November 2016 with multiple masses in her left breast. She had no history of anorexia, fever, weight loss, skin ulceration, or nipple discharge. Furthermore, the patient has no siblings, and her family history was insignificant except for diabetes mellitus. On clinical examination, five well-defined mobile masses measuring 9, 8, 5, 3, and 2 cm were present. She did not have axillary lymphadenopathy, and the results of her blood examinations were within normal limits. The breast masses were excised, and microscopically, sections revealed a diffuse proliferation of fibrous tissue, surrounding acinar and cystically dilated ductal structures with no evidence of malignancy within the limits of the specimens  recommends wide excision in malignant PTs, and there have not been enough studies confirming the efficacy of adjuvant radiation therapy after margin-negative surgery . HoweverThe NCCN recommends consideration of adjuvant radiation therapy for large malignant PTs if negative margins are not obtained, as in our patient’s case, and the surgical management of a local recurrence was not performed due to potential risk of morbidity . Thus, tOur patient’s very rare case represented a hard diagnostic challenge due to the rarity of presenting at a young age; difficulties in the differential diagnosis; and the combination of juvenile FAs, giant FAs, and malignant PTs with an extraordinary number of relapses. However, with clinical correlation and detailed observation of morphologic findings, we were able to confirm the diagnosis every time.


Carcinosarcoma of the gallbladder (CSGB) is an extremely rare tumour that presents variable proportions of malignant epithelial and mesenchymal elements. Preoperative diagnosis of CSGB is challenging owing to its non-specific clinical presentation and imaging findings. The final diagnosis requires histopathological confirmation of both the epithelial and mesenchymal components. Owing to the low incidence and poor prognosis of this tumour, it is essential to gather all the individual experience-based information. We report a case of a 52-year-old female who presented with right upper abdominal pain and vomiting for 2 weeks and a painless palpable mass on the right upper quadrant of the abdomen. Imaging studies showed a complex gallbladder mass, which was pathologically confirmed to be CSGB. To our knowledge, this is the first CSGB presented from a radiological perspective. A short literature revision of CSGB is provided. Carcinosarcoma of the gallbladder (CSGB) is an extremely rare tumour that presents variable proportions of malignant epithelial and mesenchymal elements. Preoperative diagnosis of CSGB is challenging owing to its non-specific clinical presentation and imaging findings. The final diagnosis requires histopathological confirmation of both epithelial and mesenchymal components. Owing to the low incidence and poor prognosis of this tumour, it is essential to gather all the individual experience-based information. We report a case of a 52-year-old female who presented with right upper abdominal pain and vomiting for 2 weeks and a painless palpable mass on the right upper quadrant of the abdomen. Imaging studies showed a complex gallbladder mass, which was pathologically confirmed to be CSGB. To our knowledge, this is the first CSGB presented from a radiological perspective. A short literature review of CSGB is provided.A 52-year-old female presented to the emergency department with right upper abdominal discomfort and vomiting for 2 weeks. Her personal and family history were unremarkable. On physical examination, there was a palpable painless hard mass on the right upper quadrant of the abdomen. The remaining physical examination was unremarkable.−1 ; aspartate transaminase level, 41 U l−1 ]. The remaining laboratory tests were normal, including total bilirubin and alkaline phosphatase values, as well as tumour markers—carcinoembryonic antigen, cancer antigen (CA) 19-9, CA 125 and α-fetoprotein. Chest X-ray was unremarkable.Serum liver enzyme levels were slightly elevated  and CSGB, were considered and are discussed ahead (Discussion section).i.e., adrenal carcinoma) or metastasis, could not be ruled out as there was no definite identification of the right adrenal gland. It would be unlikely for a primary adrenal malignancy to extend up to the gallbladder without invasion of any other organ, especially the kidneys. Furthermore, primary adrenal carcinoma usually manifests as a large mass, frequently with areas of necrosis and haemorrhage, which were not seen in our case, and sometimes with variable lipid content. Moreover, distant metastases are frequently found at presentation. On the other hand, adrenal metastasis was unlikely in a patient without a known primary malignancy or any other metastatic involvement. Additionally, the more common adrenal metastases usually manifest as solid lesions with central necrosis, which was not seen in our case.Adrenal origin, including adrenal primary malignancy ,4 first described by Karl Landsteiner in 1907.5 The diagnosis of CSGB relies on the concomitant demonstration of variable proportions of malignant epithelial (carcinoma) and mesenchymal (sarcoma) elements.7 The occurrence of this malignancy in other organs, such as the uterus, lungs, oesophagus and kidneys, is rare but more frequent than in the gallbladder.4Gallbladder cancer is relatively rare in the USA and most western European countries, with an incidence of approximately 1.2 per 100,000 persons, the vast majority being adenocarcinomas.2 It is common for infiltrative tumours to invade the subserosal plane into the liver. On the other hand, papillary tumours usually show a polypoid cauliflower-like appearance that tends to fill or replace the gallbladder lumen and may be present in 40–65% of cases.2 In 15–25% of cases, the initial detection of GBC appears as an intraluminal polypoid lesion with or without a thickened implantation base.2 In our case, CSGB showed no macroscopic tumour infiltration into the liver and, despite the presence of polypoid lesions, the mass was essentially exophytic, not filling or replacing the gallbladder lumen. Interestingly, in a series report of 17 uterine carcinosarcomas, 53% of cases showed exophytic growth, while none showed invasive growth.8 On MRI, CSGB appeared iso- to hypointense on T1 weighted images, without areas of high signal intensity that could be related with intralesional haemorrhage; and moderate heterogeneous hyperintensity on T2 weighted images. The moderately high signal on T2 weighted images, with a heterogeneous septated appearance, might be the strongest discriminator, as it resembles the appearance of visceral sarcomas, such as liver sarcomas,9 which could be regarded as a relatively distinctive feature of these tumours owing to its mesenchymal component. This high T2 signal intensity is very unlikely to be seen in GBC, which tends to appear with a more homogeneous and intermediate-to-low T2 signal intensity.10 Tanaka et al8 showed that, in 88% patients with uterine carcinosarcomas, more than half of the tumours showed higher signal intensity than the outer myometrium, which usually demonstrates an intermediate T2 signal intensity.8 The identification of speckled calcification within the tumour may also suggest the diagnosis of ossifying carcinosarcoma.5 In our case, despite cartilaginous differentiation, calcifications were not seen on the MDCT scan. Moreover, large GBC may show hypervascular rim enhancement on the arterial dominant phase images,3 which was not seen in our case.Preoperative diagnosis of CSGB is very difficult by imaging. Radiology literature describing the appearance of CSGB is remarkably scarce. It appears that CSGB is not associated with specific radiological signs. The main differential diagnosis is GBC, which tends to present in infiltrative or papillary forms. The infiltrative pattern may present as focal or diffuse asymmetric wall thickening in 20–30% of cases.6 In the majority of carcinosarcomas, the epithelial component consists of adenocarcinoma, and less often squamous cell carcinoma. The mesenchymal component consists of undifferentiated stellate and spindle-shaped cells that are occasionally accompanied by various proportions of heterogeneous elements, including chondrosarcoma, osteosarcoma, rhabdomyosarcoma and leiomyosarcoma.6The final diagnosis requires pathological evaluation, including immunohistological staining demonstrating the epithelial component as positive for cytokeratin and the mesenchymal component as positive for vimentin.11 CSGB usually presents with non-specific symptoms and signs, similar to those of GBC, including right upper abdominal pain, loss of appetite, weight loss, general fatigue, jaundice and vomiting. Tumour markers such as α-fetoprotein, carcinoembryonic antigen and CA19-9 are non-specific.7Similar to GBC, CSGB shows a female predominance with reported mean age at diagnosis of 66.5–72 years.7 The prognosis is poor owing to the usual presence of locally advanced disease at diagnosis, with a reported mean survival of 2.9–6 months in general, and a 1- and 5-year survival rate of 19 ± 5% and 16 ± 5%, respectively.5 Those who present with symptoms suggestive of acute cholecystitis usually have early stage disease and a better prognosis, with a 5-year survival rate of 88.9% after curative resection, when the invasion is restricted to the muscularis propria.5CSGB is a very aggressive malignancy that usually presents with large size and spreads by invading the adjacent organs, and haematogenous and lymph node metastases. Regional, retroperitoneal and para-aortic lymph nodes may be involved.11Some studies suggest tumour size as a major prognostic factor . They also indicated that the presence of gallstones, epithelial and mesenchymal component types, age and sex were of little prognostic value.7 Recently, adjuvant chemotherapy using new drug agents has been considered as the standard therapeutic option following surgical resection.7Owing to the sparse literature and poor prognosis, therapeutic interventions have not been well defined yet. Surgical resection remains the first line of management but the poor surgical outcomes and the fact that conventional chemo- and radiotherapy have not proven to be successful may suggest that new adjuvant strategies are warranted.The low incidence and poor prognosis of this tumour makes it essential to gather all the individual experience-based information in an attempt to define possible radiological features that may suggest preoperative diagnosis. MRI may be the imaging modality of choice, as the mesenchymal component may show relative distinctive characteristics, unlikely to be present in other gallbladder tumour types. In some cases, the detection of speckled calcifications on MDCT scans may also suggest the correct diagnosis.Carcinosarcomas of the gallbladder are rare tumours that present variable proportions of malignant epithelial and mesenchymal elements.Gallbladder carcinosarcoma usually presents with non-specific symptoms and signs, similar to those of adenocarcinoma.Gallbladder carcinosarcoma usually presents as a large mass. The mesenchymal component may show distinctive characteristics on MRI.In some cases, the detection of speckled calcifications on MDCT scan may suggest the diagnosis.Surgical resection remains the first line of management, with poor prognosis owing to the aggressive behaviour of the tumour and the presence of locally advanced disease at diagnosis.As malignancy tends to spread immediately to the abdomen, a different surgical technique may be required to improve the prognosis.Informed consent to publish this case (including images and data) was obtained and is held on record.


Schistosoma mansoni.Small bowel obstruction (SBO) is a common surgical condition, while Meckel’s diverticulum (MD) is the most common congenital anomaly of the gastrointestinal (GI) tract, for which differential diagnosis is considered among children. Intestinal parasitosis refers to a group of disease caused by one or more species of protozoa, cestodes, trematodes, and nematodes. Intestinal parasitic infections (IPIs) caused by pathogenic helminths and protozoan species are endemic throughout the world. The intestine is frequently involved during Schistosoma infection, especially with Schistosoma species was initiated.A 41 years old Egyptian male with an unremarkable past medical or surgical history presented to the emergency department with two days history of abdominal pain associated with nausea, vomiting, and abdominal distention. The pain was colicky and started gradually without any bowel motion or flatus for two days. Patient had tachycardia with other vitals within the normal limit. The abdomen was found to be distended and tender all over with an empty rectum by digital rectal examination (DRE). Upright chest and abdominal X-rays showed multiple air fluid levels. CT abdomen showed dilated proximal small bowel loop and a picture of SBO at distal ileal loops. The patient was resuscitated with IV fluids and shifted to the operating room. Laparoscopic exploration showed dilated small bowel loops with collapsed distal ileum, and an inflammatory band was found 20 cm from ileocecal junction, originating from a small bowel diverticulum. The inflammatory band was released, and the diverticulum was resected at the base by gastrointestinal anastomosis (GIA). Postoperatively, the patient significantly improved, passed bowel motion and was discharged for home. The histopathology report showed MD with Schistosoma infection. The patient was then referred to the infectious disease department and praziquantel, a drug of choice for schistosomiasis, effective against all Schistosomiasis is a parasitic disease that is transmitted through contact with contaminated water sources. Our patient had complete SBO with significant symptoms. He was treated with the surgical approach with diverticulum resection. Intestinal schistosomiasis was found on histopathology testing. In summary, we report a rare presentation of acute small bowel obstruction with an inflammatory band around MD harboring parasitic infection.Schistosoma mansoni infection.Diagnostic laparoscopy should be considered for all patients having virgin abdomen presenting with complete bowel obstruction. To the best of our knowledge from the literature review, this is the first case to be reported as acute SBO secondary to MD harbouring  We report a rare presentation of acute SBO with an inflammatory band around MD. The work has been reported in line with the Consensus Surgical CAse REport (SCARE) criteria [Small bowel obstruction (SBO) is a common surgical condition, accounting for 50 % of emergency laparotomies each year in the UK and over 300,000 admissions in the United States of America (USA), annually. The condition is associated with a mortality rate of approximately 10 %, and high morbidity rates among survivors. The causes of SBO are diverse; and surgery, may be indicated, depending on the suspected cause and patient factors . Most pa2Schistosoma mansoni. The patient was referred afterwards to the infectious disease department for initiation of praziquantel, which is the drug of choice for schistosomiasis and is effective against all Schistosoma species. This is an extremely rare case of acute SBO. It demonstrates the importance of diagnostic laparoscopy in all patients with virgin abdomen who present with acute bowel obstruction.A 41 years old Egyptian male with an unremarkable past medical or surgical history presented to the King Saud medical city emergency department, with two days history of abdominal pain, associated with nausea, vomiting, and abdominal distention. The pain was colicky and started gradually without any bowel motion or flatus for two days. The drug and family history was unremarkable. Patient had tachycardia, with normal blood pressure and oxygen saturation of 100 % in room air. The abdomen was found to be distended and tender all over with an empty rectum by digital rectal examination (DRE). Upright chest and abdominal X-ray showed multiple air fluid levels with no air under the diaphragm . CT abdo3MD originates from incomplete obliteration of the vitelline duct, which occurs around the fifth week of gestation. The complications of MD are greatest in the first two years of life. Haemorrhage, secondary to peptic ulceration, is the most frequent manifestation. Next, is the frequency of intestinal obstruction and intussusception being the leading cause. Intestinal obstruction accounts for 30%–56% of symptoms of MD. Commonly documented mechanisms of obstruction are volvulus, intussusception, bands, Littre's hernia, internal hernias, and strictures .Schistosoma mansoni and Schistosoma japonica represent the types that infest the intestine [Schistosomiasis is a parasitic disease transmitted through contact with contaminated water sources. It is related to bad personal hygiene and environmental sanitation. This explains the dominance of this disease in the endemic areas within the developing countries. The disease is endemic in Sudan and Egypt. ntestine .Schistosoma species. In summary, we report a rare presentation of acute small bowel obstruction with an inflammatory band around MD harbouring parasitic infection. To the best of our knowledge from the literature review, this is the first case report to be reported on acute SBO secondary to MD harbouring Schistosoma mansoni infection.Our patient had complete SBO with significant symptoms. He was treated with laparoscopic exploration with band release and diverticulum resection. Intestinal schistosomiasis was found on histopathology. He was treated with praziquantel after surgery, which is the drug of choice for schistosomiasis and is effective against all No fund to my research to be disclosed.This is case report study and ethical approval not required.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.Dr.Emad Aljohani (case description and discussion).Dr.Fahad Almadi (literature review and collected the data from the patient file).5143.Dr. Emad Aljohani.Not commissioned, externally peer-reviewed.No conflict of interest.


Abdominal wall endometriomas are quite uncommon. They are usually misdiagnosed by both the surgeon and the gynaecologist. Awareness of the details of this rare condition is therefore essential for prompt diagnosis and adequate treatment. Endometriosis though a condition commonly seen in the pelvic region can also occur at extrapelvic sites giving rise to a diagnostic dilemma. Abdominal wall endometrioma is one such complex variant of extrapelvic endometriosis with an incidence of less than 2% following gynaecologic operations. A case of abdominal wall endometrioma diagnosed clinically and treated by wide surgical resection is presented to highlight the importance of clinical evaluation in the diagnosis of this condition. The etiopathogenesis, presentation, investigations, and management are discussed briefly. Clinical evaluation confirmed by supportive imaging is diagnostic. Wide local excision is the mainstay of treatment. Endometriosis is defined as a benign inflammatory disease characterized by the presence of ectopic endometrial tissue which is oestrogen dependent. When the lesion is a well circumscribed mass it is designated as an endometrioma. Abdominal wall endometrioma (AWE) is an uncommon aftermath of gynaecologic operations such as caesarean section or an abdominal hysterectomy. The incidence varies from 1 to 2% [A 29-year-old female presented with a mass on the anterior abdominal wall present for 1 year. The mass had gradually increased in size over this period of time. She complained of continuous discomfort and pain which became worse during menses. The mass increased in size during menses as per the patient's description. She had undergone a caesarean section six years back. There was no other significant history. Her menses were regular with no bladder or bowel disturbances.Physical examination of the abdomen revealed a circumscribed mass measuring approximately 6 cms in diameter in the infraumbilical region to the left of the midline .The mobility of the swelling became restricted on contracting the underlying muscles suggestive of infiltration of the underlying musculoaponeurotic structures.Laboratory investigations were within normal limits. A contrast enhanced CT scan was done during her menses. The CT scan revealed a contrast enhancing lesion in the subcutaneous tissues infiltrating the underlying musculoaponeurotic structures highly suggestive of an abdominal wall endometrioma .She underwent surgical resection. The endometrioma was resected along with the portion of the underlying aponeurosis and rectus abdominis muscle Figures .A polypropylene mesh was placed over the defect created by the resection and fixed all around the defect to the anterior rectus sheath with nonabsorbable sutures . The posFollowing the procedure there was no seroma at the operative site and the patient noted complete relief of symptoms.The presence of endometrial glandular and stromal tissue outside the uterus is called endometriosis. It is seen in women of active reproductive age , 2. The Various theories have been proposed to explain the etiopathogenesis of abdominal wall endometriomas . The traPrevious caesarean section or hysterectomy, high parity, and increased menstrual flow are known risk factors for AWE. The Esquivel triad comprised of a palpable tumour, cyclic pain, and a history of lower caesarean section is virtually diagnostic of AWE .However this may not be the presentation in all cases. Subtle variation in clinical features may be seen. Hence an elaborate history with respect to time frames of surgical events and commencement of symptoms is extremely important. Usually the time interval between the index surgery and onset of symptoms ranges between three and six years. In the case presented it was almost six years. The diagnostic clue is exacerbation of local symptoms of severe pain and discomfort accompanied by expansion of the mass with the onset of menstruation. This was typically seen in the case presented thus enabling an accurate clinical diagnosis.A variety of imaging modalities are available to confirm the diagnosis of AWE. The surgeon has to be aware of the findings revealed by each modality in order to avoid misdiagnosis. Doppler ultrasound typically reveals a solid hypoechoic lesion containing internal vascularity. It has a sensitivity of 92% –6. ContrFine needle aspiration cytology (FNAC) runs the risk of needle track implantation of the endometriotic lesion. FNAC will show endometrial-like epithelial cells, stromal cells, and hemosiderin laden macrophages. It is extremely difficult to diagnose scar endometriosis by FNAC .Histopathological evaluation of the resected mass is confirmatory. Any two out of the three classical features are diagnostic. These include endometrial glands, endometrial stroma, and hemosiderin laden macrophages.The risk of developing a clear cell carcinoma in these lesions is less than 1%. Advanced age, postmenopausal state, and tumour size greater than 9 cms are risk factors for malignant transformation. The five-year survival rate in such cases is 80%.The differential diagnosis may include a variety of conditions such as hernia, lipoma, desmoid tumour, or primary or metastatic malignancy. Therefore a careful history with proper interpretation of radiological findings can help in making a correct preoperative diagnosis.Various pharmacologic treatments have been used for AWE , 11. TheSurgery consists of wide local excision with an aim of achieving a clear resection margin . The undAbdominal wall endometrioma is a rare entity developing after gynaecologic surgery. Awareness of this condition is essential for making a diagnosis. A proper history and physical examination during menses can help in arriving at a diagnosis. CECT will confirm the diagnosis. Wide local excision is the mainstay of treatment. High pressure saline irrigation of the wound edges can prevent the development of AWE at the time of the index surgery.


Ruptured cornual abscess or pyometra can resemble other more common causes of acute abdomen, including appendicitis, diverticulitis, tubo‐ovarian abscess, and perforated viscus. Despite its rarity, the diagnosis of ruptured pyometra should always be considered in females presenting with acute abdominal pain, particularly in the setting of a retained intrauterine device. Question: What is the diagnosis?A 42‐year‐old female presented with acute abdominal pain and imaging findings shown in Figure Answer: Exploratory laparotomy revealed a ruptured right cornual abscess Figure .Pyometra, a uterine infection, generally occurs in postmenopausal women in the setting of gynecologic malignancy.2 Management also becomes increasingly complex, as septic shock may ensue with abscess rupture, necessitating intensive care and prolonged hospitalization. Infection can recur/persist in up to 33% of casesDiffuse peritonitis resulting from ruptured pyometra can be observed in other gynecologic  and non‐gynecologic  conditions. Imaging can help delineate the source; however, in this case, imaging was equivocal for TOA vs appendicitis, making definitive diagnosis difficult. Prompt diagnosis of pyometra is critical, as mortality approaches 25% with rupture.None declared.LL/PP: participated in the care of the patient, manuscript writing and editing, and literature review. BB: participated in manuscript writing and editing. MF: participated in the care of the patient and literature review. PP: participated in the care of the patient and supervised manuscript preparation.


Spinal metastases of gastrointestinal stromal tumors are rare; however, the incidence has been increasing since the introduction of tyrosine kinase inhibitors, which have improved overall survival. Due to the rarity of cases, there are no treatment guidelines for spinal metastases of gastrointestinal stromal tumors. We describe a patient who underwent spinal metastasectomy for a rectal gastrointestinal stromal tumor; we further provide a review of all cases of gastrointestinal stromal tumors with spinal metastases.A 51-year-old Japanese man who had undergone resection for a rectal gastrointestinal stromal tumor was diagnosed with L3 vertebral metastasis 10 years after surgery. As there were no metastases to vital organs, an en bloc corpectomy of the L3 vertebral body, using bilateral retroperitoneal approaches, was performed to achieve local cure and to prevent neural compression. A 3-year follow-up showed no local recurrence or new metastases; he had full neurological function.Spinal metastasectomy can be an effective treatment for solitary spinal metastases of gastrointestinal stromal tumors. Gastrointestinal stromal tumors (GISTs) are the most common mesenchymal tumors of the digestive tract and are derived from the interstitial cells of Cajal . Spinal This is the first report of radical metastasectomy for solitary spinal metastasis of GIST.A 51-year-old Japanese man who was born and raised in Japan and did not have any underlying medical condition presented with a rectal mass. He underwent endoscopic biopsy of the lesion, and the histopathological report showed spindle-shaped tumor cells with mild cytological atypia. Immunohistochemical analysis revealed that the tumor was positive for CD117 (c-kit) and CD34, leading to the diagnosis of a rectal GIST. He underwent colectomy after shrinking the tumor with 400 mg/day of imatinib for 9 months. After completion of treatment, annual follow-up computed tomography (CT) scans showed no local recurrence or distant internal organ metastases. His postoperative course was good and he could continue working as a judo therapist. There was nothing notable in his medical history, except for the rectal GIST, and he did not take any medication after surgery. His family history was uneventful and he did not smoke tobacco or drink alcohol.Although he was asymptomatic, a CT scan 10 years after surgery revealed a destructive osteolytic lesion in the L3 vertebral body  and a C-reactive protein (CRP) level of 114 mg/L (reference range < 3 mg/L). His liver enzymes were elevated: alanine aminotransferase (ALT) 112 U/L (reference range < 35 U/L) and aspartate aminotransferase (AST) 40 U/L (reference range < 35 U/L). CRP, ALT, and AST levels completely normalized within 1 month after surgery. In repeated laboratory tests, CBC, CRP, and liver enzymes remained normal until the final follow-up. At the 36-month follow-up, no local recurrence, new metastasis, or instrumentation failure was observed on CT or magnetic resonance imaging. He had full neurological functions without any limitation of daily activities.This is a case report of metastasectomy performed for spinal metastasis, which appeared 10 years after rectal GIST surgery, with a good postoperative course. The unique points in this case are that the metastasis occurred after a very long period of time, and this is the first report of radical surgery for spinal metastasis of GIST.KIT (78%) or PDGFRA (6%) genes [GISTs are spindle, epithelioid, or pleomorphic mesenchymal tumors of the digestive tract that express CD117 (c-kit) [%) genes . Even in%) genes , 5, and r PDGFRA % genes [Bone metastases of GISTs, including the spine, are rare and only 12 cases with spinal metastases have been described to date  of GIST and benefitted from long-term disease-free survival [Few data are available in the literature on the treatment of spinal metastases in GISTs. Previously, most cases of spinal metastases were treated non-surgically with TKIs with or without zoledronic acid. Pain reduction and disease stabilization were observed in some cases, although the mechanism remains unclear , 9, 11. survival . AlthougIn conclusion, although the incidence of spinal metastasis of GISTs is currently low, it could become more prevalent because of longer patient survival after TKI therapy, which offers sufficient time for the occurrence of metastasis. The possibility of late metastasis, such as in the present case, should be acknowledged and more attention should be paid to the diagnosis of spinal metastases of GISTs. From the satisfactory outcome of this case, spinal metastasectomy can be an effective treatment for solitary spinal metastasis of GISTs. Furthermore, more case series should be extensively studied to establish surveillance protocols and treatment guidelines.


Introduction: We present a case of a gentleman with a new diagnosis of intracranial plasma cell myeloma. PET scan as part of work-up identified the incidental presence of increased uptake primarily of the large vessel arterial system consistent with vasculitis. Review of the literature describes association of vasculitis and haematological malignancy (such as myelodysplastic syndrome and lymphoprolferative disorders), however the association with myeloma  appears to be less frequent with limited reported cases. We therefore wish to highlight this case for the interest of our rheumatological colleagues.Case description: A 71 year old gentleman presented acutely with worsening vertigo symptoms and associated nausea. He had been experiencing such symptoms over the preceding two months, with his condition gradually progressing over that time. He had been experiencing intermittent right-sided occipital headaches, and following review from his GP during this period, he was started on gabapentin to manage a provisional diagnosis of occipital neuralgia. Apart from the above, he denied systemic features of weight loss, pyrexia, skin rash, myalgia or arthralgia. The patient had an extensive past medical history including monoclonal gammopathy of undetermined significance (IgG lambda paraprotein), IgG-mediated peripheral neuropathy, postural hypotension and previous diagnosis of polymyalgia rheumatica (in remission at time of presentation and not on therapy). Despite the above co-morbidities, prior to his recent symptoms, this gentleman was otherwise fit and well, and was only under observation in regards to his MGUS diagnosis via the local haematology team. On presentation, no obvious objective clinical abnormality could be identified including neurological deficit. GCS was 15/15. Routine blood tests were within normal range including inflammatory markers of CRP and ESR. In view of progressive vertigo symptoms, concerns of a possible intracranial lesion was considered, and as such arrangements were made for an urgent MRI head scan. This was reported to show an isointense, homogeneously enhancing destructive mass centred on the right occipital skull base, with a significant extraosseous component indenting the right cerebellar hemisphere with gross oedema. The lesion was approximately 6 cm in size. No hydrocephalus was present at time of the scan. Urgent neurosurgical opinion was advised and dexamethasone therapy (8 mg BD) was initiated. Neurosurgical review was arranged at the local centre, and biopsy of the lesion was performed. Histology demonstrated tissue diffusely infiltrated by plasma cells positive for CD138, CD79a and monotypic for lambda light chain - features were deemed consistent with plasma cell myeloma. Bone marrow trephine reported a small population of lambda light chain predominant plasma cells, confirmed by CD-138 potentially suggestive of myeloma. CT chest/abdomen/pelvis did not identify any obvious primary/secondary lesions. PET CT scan was arranged, which as well as the intrancranial lesion described, also reported generalised FDG uptake of the aorta, subclavian, axillary, common/external iliac and femoral arteries - findings were reported as consistent with large vessel vasculitis and were identified despite dexamethasone use. His case was referred to the haematology team for further management and decision has been made for radiotherapy treatment of the intracranial lesion. Incidental vasculitic changes described on PET scan have been attributed as secondary to his haematological condition, and arrangements are made for re-imaging following treatment.Discussion: Associations between malignancy and vasculitis have been previously reported within the literature, with approximately 5-8% of vasculitis cases having associated malignancy. Reports suggest that haematological malignancies are more commonly associated than organ solid tumours, with sub-types such as MDS and lymphoma being most prominent. Myeloma related vasculitis appears to be less common, with limited described case reports. Various theories are made regarding the link of malignancy and vasculitis, with cause and affect between the two being unclear. For example, one potential association with myeloma is the development IL-6 genes resulting in leucocytoclastic vasculitis (LCV). LCV has been reported among the most common vasculitides associated with malignancy, however this is usually associated with small vessel cutaneous vasculitis. Indeed, large vessel vasculitis (as suggested by this case) related to myeloma appears to be extremely rare based on limited reports available. As per our described case, the patient did not appear to have findings suggestive of systemic vasculitis, and despite apparent extensive vascular involvement as per PET imaging, inflammatory markers remained normal throughout the presentation (even prior to dexamethasone usage) and appears to be an incidental finding from imaging. As mentioned, our patient is currently undergoing treatment for his primary intracranial lesion under the haematology team. Following therapy, it would be interesting to review whether apparent PET findings settle once the primary myeloma is managed.Key Learning Points: The primary presentation for this patient focussed on his symptoms of persistent vertigo and associated nausea. Through extensive investigations involving multiple specialties, we were able to identify the causative lesion and histology suggestive of intracranial myeloma. Incidental finding of large vessel vasculitis on PET scan has highlighted a potential association between the two diagnoses. Myeloma and large vessel vasculitis appears to be rarely reported within the literature, and as such we feel this particular case may be of interest to the rheumatological community. Follow up on completion of myeloma treatment is planned to review possible resolution of PET findings.Disclosure: S. El-Ghazali: None. K. Bhamra: None. A. Khan: None. M. Burden: None. V. Nallapuneni: None. I. Patel: None.


Primary leiomyosarcomas of vascular origin are rare tumors. They frequently arise within the inferior vena cava; however, the peripheral vein was also affected. To date, only a few hundred cases have been reported in the world literature. Although it is an extremely aggressive tumor, the symptoms may be unspecific, especially in the lower extremities. In this report, we present a case of primary vascular leiomyosarcoma, arising from the short saphenous vein, with symptoms mimicking thrombus in the initial diagnosis. The diagnosis of leiomyosarcomas was confirmed by standard H&E staining and immunohistochemical staining. Recurrence of the tumor has been observed five years after surgical treatment. Due to its rarity, experience in the management of this type of tumor is limited. The mainstay of treatment for these tumors is complete surgical resection. The purpose of the presented case is to discuss the clinicopathological features and management options of this tumor, under the light of the most recent literatures. Primary tumors arising from the vessel wall are rare. Although primary vascular leiomyosarcoma (vLMS) arising from larger blood vessels (particularly in the inferior vena cava and large veins of the lower extremity) has been reported , leiomyoA 58-year-old woman was admitted to a cardiovascular surgery clinic for the evaluation of a painless mass in her left leg and lower extremity edema with a three-week duration of history in August 2008. Physical examination showed a mass was palpable deep to the skin in the course of the short saphenous vein. No other skin lesions and no systemic manifestations were found. Routine laboratory studies were unremarkable. Ultrasonography detected a luminal mass in the short saphenous vein. A magnetic resonance imaging (MRI) revealed a 15 × 10 × 7 mm heterogeneous mass extending into the Achilles tendon, which may be related to the thrombosis of the short saphenous vein. The mass was excised en bloc under tourniquet control with a safe margin. Vascular reconstruction was not performed. The gross specimen consisted of the saphenous vein was surrounded and infiltrated by a whitish to brownish, rubbery tissue all along its course, measuring 3.5 × 2 × 1.5 cm. Microscopic examination showed a tumor was arising from the vascular wall  and compLeiomyosarcomas of soft tissue generally present retroperitoneum; however, it is the predominant sarcoma arising from blood vessels. vLMS more commonly arises from the venous, rather than arterial vessels. It is reported that the long saphenous vein is the most affected vein and followed by the femoral vein and popliteal vein in the lower extremity . On the The symptoms are related to the location of the tumor, rate of tumor growth, and degree of collateral blood flow. They most commonly present as slow-growing, painless masses. When the tumor is occlusive, the symptoms can mimic those of deep vein thrombosis , 7. In aHistological tumor was composed of spindle-shaped cells with an eosinophilic cytoplasm with muscular striation and cigar-shaped rounded nuclei. We applied immunohistochemical staining for smooth muscle markers such as actin and desmin as well as h-caldesmon to verify the diagnosis. The differential diagnosis includes spindle cell-shaped neoplasms such as benign and malignant tumors of the nerve sheaths, myofibroblastic tumors , synovial sarcoma, and fibrosarcoma.Leiomyosarcomas have clinical and biologic differences according to site-related subgroups. Although leiomyosarcomas of the retroperitoneum and abdominal cavity are associated with an aggressive clinical course, the behavior of the primary vLMS has been controversial. Leu and Makek reported that small vLMSs might have relatively good prognosis ; howeverOperation is the mainstay of treatment for vLMSs. The role of neo-adjuvant chemotherapy (CT) and radiotherapy (RT) in the management of disease is still unknown. The rate of local recurrence was not significantly different between patients who received perioperative RT and those who did not . SimilarAlthough there has been local recurrence in the five-year period, the present case seems to be a good prognosis. The review revealed that the survival of the deceased patients with the vLMS of the popliteal artery was between 7 months and 4 years . Reix etIn conclusion, vLMSs of extremity are rare and aggressive tumors. Clinicians should consider that it may be misdiagnosed as deep vein thrombosis in the initial diagnosis. The management of these tumors is surgical treatment; however, considering the local recurrence in our patient, radiation therapy and chemotherapy should have been considered. Due to its rare incidence, all publications in the literature are of small clinical series or case reports. Therefore, we thought that our case report also provides data to the literature for the management of vLMS.


Mycobacterium shimoidei infection in an immunosuppressed 61‐year‐old male with a background of emphysema. His chief complaint was haemoptysis. Chest computed tomography showed a large, thin‐walled cavitary lesion in the upper right lobe. Although NTM were identified in two separate expectorated sputum samples, DNA–DNA hybridization (DDH) failed to identify the species. M. shimoidei was finally identified using matrix‐assisted laser desorption/ionization time‐of‐flight mass spectrometry (MALDI‐TOF MS). Following antimicrobial agent susceptibility tests, treatment with clarithromycin, levofloxacin, and ethambutol commenced. Six months post‐treatment, acid‐fast sputum culture was negative and repeat imaging demonstrated improvement of the radiographic abnormalities. This study aimed to assess the utility of MALDI‐TOF MS for successful identification of rare NTM species that are not identifiable by DDH. It is the first report of M. shimoidei from Okinawa, which is the only prefecture in Japan categorized as subtropical.Non‐tuberculous mycobacteria (NTM) cause several infectious diseases in humans. This study reports on  Mycobacterium shimoidei is a slow‐growing NTM that was first isolated in Japan in 1968, gaining species status in 1975 Non‐tuberculous mycobacteria (NTM) are ubiquitous in the environment and can cause various infectious diseases in humans. The prevalence of NTM‐induced lung infections is increasing globally. A 61‐year‐old man from Okinawa, Japan, presented at his local hospital complaining of cough and bloody sputum during the preceding week. He was a current smoker with a history of chronic obstructive pulmonary disease (COPD), and rheumatoid arthritis that had been treated with prednisolone 7.5 mg per day and methotrexate 12 mg per day since 2005 by a primary care physician. He was a professional gardener. Chest X‐ray demonstrated a thin‐walled cavitary lesion in the upper right lobe. He was referred to our hospital for further evaluation. Chest computed tomography (CT) on the first visit showed a large, thin‐walled cavitary lesion, pleural wall thickening, trabecular and linear shadows in the upper right lobe, and bronchiectasis in the upper and middle lobe , with the breakpoints derived using Mycobacterium kansasii as a reference. M. shimoidei proved susceptible to clarithromycin (CAM), ethambutol (EB), streptomycin, amikacin, and levofloxacin (LVFX); and resistant to rifampicin (RFP). After referring to published literature and the advice of an expert on acid‐fast bacteria, the patient was started on a treatment regimen of CAM, EB, and LVFX in January 2018. His haemoptysis decreased gradually and the cavitary lesion improved , and M. shimoidei that is not identifiable by DDH. This is essential for choice of the appropriate antimicrobial treatment. Notably, this is also the first identification of M. shimoidei in Okinawa, the southernmost and only prefecture categorized as subtropical in Japan. Species isolated from patients with NTM lung disease are geographically diverse, although the Mycobacterium avium complex is the commonest species in most countries, including mainland Japan Mycobacterium abscessus complex is the predominant pathogen causing NTM lung disease The purpose of this study is to communicate the potential utility of MALDI‐TOF MS for identification of M. shimoidei infections have been predominantly in the lungs, and largely among male patients. The most commonly associated concurrent conditions were COPD, bronchiectasis, and past TB Previously published details of 25 cases and this case are summarized together in Table MALDI‐TOF MS is a proteomics method that permits rapid and accurate identification of mycobacterium species from positive cultures M. shimoidei is required, it is known to be resistant to isoniazid and RFP, and susceptible to EB and rifabutin (RFB) M. shimoidei. Increased recognition and understanding of this pathogen are necessary to expedite diagnosis and improve patient outcomes.Although more information about drug susceptibilities of Appropriate written informed consent was obtained for publication of this case report and accompanying images.