A 2‐year‐old girl developed three mucoceles on lower lip, probably due to her habit of rolling lower lip behind the maxillary anterior teeth. The spacing in the dentition and mechanical irritation from habitual lip biting may have caused the mucoceles. At 3 years after excision surgery, the patient remained recurrence‐free. Most mucoceles develop as a solitary lesion, and only a few cases of multiple lesions have been reported.2Our patient was a 2‐year‐old female infant. She presented with three painless vesicles on the lower lip that had emerged 3 weeks earlier. The vesicles had been left untreated due to their fluctuation in size. However, a local dentist who saw the patient for a routine checkup recommended a thorough examination of the vesicles, and thus referred her to our hospital. Her medical and family histories were unremarkable. At the initial visit, three well‐circumscribed round masses, each measuring approximately 5 mm in diameter, were found on the lower lip. The lesions were bluish fluctuant masses covered with normal mucosa. No pain or congestion was noted. The facial appearance was symmetric without any skin rashes. No swelling or tenderness was present in the submandibular or cervical lymph nodes. The patient had no fever and her food intake was good. The differential diagnosis of mass lesions in lower lip is salivary gland tumors, fibroma, and hemangioma. Salivary gland tumors of the minor glands can occur on the lip but are more commonly seen in the upper lip rather than in the lower lip. Fibroma is commonly pink and nonfluctunt. Hemangioma can undergo transient reduction in size under pressure. Based on these findings, the lesions were diagnosed as multiple mucoceles of the lower lip. The mucoceles were individually excised, along with the surrounding minor salivary glands, under local anesthesia. We removed three spherical masses, each measuring 5 × 5 × 5 mm in size Figure . HistopaHer postoperative course was uneventful without any infection or wound dehiscence. To date, 3 years after the operation, the patient has not experienced any recurrence. Informed consent was obtained from the patient's parents, and the procedures were performed in accordance with the Helsinki Declaration.3A mucoceles is the most common type of cyst that occurs in oral soft tissues. Chi et alThe most likely causes of mucous cysts are injuries or chronic stimulation.Histopathologically, mucoceles can be classified into (a) the extravasation type  and (b) the retention type .Mucoceles are located in submucosa and appeared as translucent small blisters covered with normal mucosa. Mucoceles can rupture due to friction from eating, and they may disappear, but then often recur. Therefore, the optimal treatment is to excise the lesions along with the surrounding minor salivary glands. As alternative treatment options, cryotherapyWhen performing excision in a patient with multiple mucoceles, as in the present case, potential lip deformities caused by suturing is a concern. However, in the present case, each cyst was small and spatially separated from one another. Therefore, the lesions were removed separately, and the wounds individually sutured, which resulted in an uneventful postoperative course without lip deformities or recurrence. The recurrence rate of mucoceles after excision is approximately 7%; many of these are of the extravasation type, as in the present case. We have continued to follow up the patient while providing guidance for improving the parafunctional habit linked to the development of the mucoceles. The clinical course of the patient has been favorable without any recurrence at the 3‐year follow‐up.4We reported a case of multiple mucoceles on the lower lip in a 2‐year‐old girl. The reason for the occurrence of multiple cysts on the lower lip in this case may have included the habit of lip biting, spaces between the primary teeth, and the density of the minor salivary glands in the lower lip.None declared.AA: developed the concept and design of the study. KK: revised critically the manuscript for important intellectual content and gave the final approval of the version to be submitted. HH and MM: drafted the article.


Most of the lesions in the oral cavity have papillary appearance. Oral squamous papilloma (SP) is one such type, which is a benign proliferation of the stratified squamous epithelium and presents as papillary or verrucous exophytic mass induced by human papillomavirus (HPV). Most of the oral mucosal lesions are often asymptomatic and have small progression. The common sites of occurrence include tongue, soft palate, and uvula. Squamous papilloma arising on hard palate is described in this article. Surgical excision of the lesion was done and sent for histopathological analyses that confirmed the clinical diagnosis. In larynx and trachea, malignant transformation of papillomas has been reported. The potentially malignant nature of SP if present needs to be explored.How to cite this article: Chaitanya P, Martha S, Punithvathy R, Reddy M. Squamous Papilloma on Hard Palate: Case Report and Literature Review. Int J Clin Pediatr Dent 2018;11(3):244-246. Oral SPs appear as verrucous growth. They present as exophytic mass in the oral cavity induced by HPV. They are benign proliferations of the stratified squamous epithelium.1 The sites of predilection include tongue, soft palate, and uvula, but any surfaces of the oral cavity can be affected.23 Squamous papillo-mas are reported frequently in children but it may affect any age group; 7 to 8% of all oral masses or growths in children comprise oral SP.The World Health Organization defines papilloma as “a range of localized hyperplastic exophytic and polypoid lesions of hyperplastic epithelium with a verrucous or cauliflower-like morphology.”4 Oral squamous lesions have clinical appearance that mimic exophytic carcinoma, verrucous carcinoma, or condy-loma accuminatum. However, SPs usually have a slow progression and are often asymptomatic and show benign character in pathological examinations.3It is the fourth most common oral mucosal lesion and is found in 4 of every 1,000 oral soft tissue lesions.This article describes a case of SP arising in an unusual location in the oral cavity.A 10-year-old girl reported with the chief complaint of growth of soft tissue mass on the palate that was present since 1 month. History regarding the growth revealed that it first started as painless slow growing soft tissue mass that gradually enlarged and associated with discomfort and occasional pain on eating and interference while biting. Past medical, dental, and personal history were noncontributory.General and extraoral examination of the patient did not reveal any significant findings. Intraoral examination revealed a lesion on anteriormost part of hard palate. The lesion was a pale, pink colored, pedunculated growth with finger-like projections of soft tissue present on right side of the rugae area of hard palate just adjacent to incisive papilla. It was approximately 1.5 cm in size, circumscribed, and not associated with bleeding  and 2.Based on these clinical features, the growth was provisionally diagnosed as papilloma. Surgical excision of the growth was done with a 1 mm margin to the depth of submucosa under local anesthesia. After excisional biopsy, specimen was fixed and stained with hematoxylin and eosin for histological analysis.Histological examination revealed papillary projections of parakeratinized stratified squamous epithelium of variable thickness with localized areas showing mild basilar hyperplasia with few koilocytes and enclosing connective tissue cores  and 4. T5 The lesions are softened/flaccid in 66.7% of cases and pedunculated in 75% of the lesions.2 Although etiology of papilloma is unknown, it is most commonly associated with HPV 6, 11, and 16.6 The HPV is a member of the papovavirus group. It is a deoxyribonucleic acid (DNA) virus containing a single molecule of double-stranded DNA. The virus is a nonenveloped icosahedral particle with diameter ranging from 45 to 55 nm and has 72 cap-someres in a skewed arrangement. Various species are distinct antigenetically sharing some common antigenic determinant. Replication of HPV occurs within the nuclei of epithelial cells in the spinous layer due to stimulation of host DNA synthesis. This induces a series of proliferative alterations that result in tumor growth. Controversy remains on viral transmission pathways in children, although the possibilities include sexual abuse, perinatal transmission, autoinoculation, heteroinoculation, and possibly indirect transmission via fomites.7Oral SPs are benign exophytic lesions presenting as papillary or verruciform proliferation. This lesion was first described by Tomes as gingival “wart” in 1848.8 most commonly as seen in the present case.The location of the lesion and clinical presentation in the present case are similar to common identifications of oral SP. The histopathological examination of the lesion confirmed the clinical diagnosis. The lesions are asymp-tomaticSquamous papillomas are traditionally divided into two types: Isolated-solitary and multiple-recurring. Although the isolated-solitary type of SP is common in adults, the clinical presentation of SP was similar to isolated-solitary type in this child. The differential diagnosis of solitary type of oral SP includes verruciform xanthoma, papillary hyperplasia, and condyloma acuminatum.8The clinical appearance of verruciform xanthoma is similar to squamous papilloma, but the lesion has predilection for the alveolar ridge and gingiva. For inflammatory papillary hyperplasia, a cause-and-effect relationship  should be evident. On comparison with papilloma, condyloma are larger, have a broader base, and would appear pink-to-red due to less keratinization. Clustered or multiple SPs suggest focal epithelial hyperplasia (Heck disease).2 The most characteristic feature of HPV infections is the presence of koilocytes. Koilocyte is an HPV-infected squamous epithelial cell. The cellular changes/cytopathic effects observed are enlargement of the nucleus , irregularity of the contour of nuclear membrane, darker than normal staining pattern in the nucleus, and the presence of perinuclear halo.The microscopic appearance of papilloma shows proliferation of the spinous cells in the form of long thin finger-like projections extending above the surface of the mucosa containing a thin connective tissue core. The connective tissue is continuous with stroma of the stalk, the body of the mass, and the surface projections. The epithelial surface may reveal hyperparakeratosis. Basilar hyperplasia and mild mitotic activity may be present occasionally. This should be considered with caution as it can mislead to a diagnosis of mild epithelial dysplasia. There have also been reports of superimposed mycotic infec-tions.2 koilocytes were demonstrated in 100% of the oral SP studied. Even though SP is a HPV-induced lesion, its infectivity is much less pronounced. Malignant transformation of papillomas in areas, such as larynx and trachea has been reported.1In a study conducted by Carneiro et al,9Treatment of choice for SP is surgical excision. Vaccines could be used as adjuvant treatment following surgery by generating an immune response. A decreased prevalence of SP may result in future, against types 6 and 11. Two vaccines have been developed: Cervarix and Gardasil that can prevent infections and precancerous lesion caused by HPV infection.Though SPs are common lesions of the oral cavity, their occurrence on the hard palate is rare, and its histopatho-logical features overlap with a number of other verru-copapillary lesions. Hence, differentiating papillomas from other lesions is important. With papillomas in other regions showing malignant transformations, the potentially malignant nature of SP if present should be explored.


Benign lumbar intradural tumors are statistically uncommon and usually present with complaints of back pain with or without radicular neurological complaints. This report involves two separate patients that were found incidentally to have large intradural tumors without any neurologic complaints. In both cases the tumors were discovered when having magnetic resonance imaging (MRI) after minor auto accidents. Neither patient had any pre-existing lumbar or neurologic complaints. The report will review the different regions and types of incidental findings commonly seen on lumbar MRI scans and the need for close follow-up in patients with incidental lesions such as tumors. Incidental radiologic findings are common and detected frequently with the use of higher definition studies such as computerized tomography (CT) and magnetic resonance imaging (MRI) ,2. LumbaCase 1The patient was a healthy 38-year-old male who was involved in a minor automobile accident and was complaining of intermittent back pain that he described as sore and stiff. His pain was 3/10 on visual analog scale. His pain was exacerbated by prolonged sitting and standing, and forward bending. He reported that there were no relieving factors. He was treated with therapy and had the MRI scan six weeks after the accident. It was originally read as a herniated L3-4 disc to the left side. The radiology report specifically stated L2 was normal. He started complaining of left leg pain, primarily in posterior thigh and calf, as well as tingling in the left leg. Because of failure to get symptomatic relief and complaints of tingling in the left leg, he was referred to a neurosurgeon. Neurologic examinations including sphincter function, reflexes, sensory and motor were normal. However, on review of the original MRI scan by the neurosurgeon, a possible intradural lesion was seen at L2 and a stat MRI with gadolinium contrast was ordered revealing a smooth, strongly homogeneously enhancing mass of 14 x 21 x 13 mm in the central and right intradural space behind the L2 vertebral body (Figure Case 2The patient was a healthy 47-year-old male who was involved in a minor automobile accident approximately five weeks prior to presentation. He was the restrained driver of a motor vehicle when it was struck on the driver’s side. He was complaining of constant low back pain that he described as sore, stiff and aching that radiated from the low back into the left hip. His pain was rated a 6-7/10 on a visual analog scale. Sitting and lying down exacerbated his symptoms. His symptoms improved with physical therapy, non-steroidal anti-inflammatory medication and rest. An MRI without gadolinium, of his lumbar spine was performed six weeks after his accident. It was read by the radiologist as a L4-5 posterior central and left paracentral disc herniation, L5-S1 broad-based disc bulge and an expansile mass in the conus medullaris and superior cauda equina. The radiologist recommended that the MRI be repeated with gadolinium contrast. The contrast MRI was compared to the previous MRI which also showed the intradural lesion and he was referred to a neurosurgeon. Neurologic examinations including sphincter function, sensory and motor were normal. His deep tendon reflex was normal except a decreased knee jerk on the right which was 1+/4. On review of the MRI with gadolinium contrast an intradural mass was seen in the conus at L1 measuring 2.4 cm x 1.5 cm with a non-enhancing irregular center and an enhancing periphery (Figure In the two cases presented, coincidentally seen within six weeks of each other, large incidental asymptomatic intradural tumors were found in MRI scans performed after minor automobile accidents. In each case the patient had either new specific minor lumbar pain or radiculopathy leading to the original radiologic study that was being performed for a suspecting lumbar disc. One patient actually did have a 5 mm acute herniated L3-4 disc, lateralized to the side of his pain and numbness but with an incidental intradural tumor at L2, originally missed on the initial MRI examination, while in the other patient the lumbar spine findings did not reveal any disc disease but an incidental mass that was totally asymptomatic within the conus medullaris.Incidental radiologic findings are defined as any abnormal finding not related to the chief complaint that was the reason for the radiologic study, however it is important that these incidental findings not only be noted by the radiologist but suggested additional films recommended and contact made to the referring physician for both clinical and radiologic follow-up ,2. LookiIn a large series of 1268 cases having lumbar MRI for suspected lumbar disc, 107 or 8.4% had incidental findings. These included in order of frequency fibrolipoma, Tarlov's Cyst and vertebral hemangiomas. There were no tumors in this series . Spinal Treatment for intradural tumors is surgical resection, either through standard laminotomy or more recently using a microsurgical interlaminar approach and more recently spinal stereotactic radiosurgery ,17-20. UThis report of two incidental lumbar intraspinal tumors highlights the importance of careful attention to incidental MRI findings. Careful review of radiologic films and being observant of any incidental findings is critical in detecting uncommon findings like tumors. When the CT or MRI is performed the focus is on 'disc' trauma and 'herniation' incidental findings must be noted by the radiologist or noted later if detected by another physician reviewing the films like in our first case. Although most incidental findings, whether intraosseous or intraspinal, are benign, stable lesions, findings suggestive of vertebral metastatic lesion, osteoporotic fractures and possible intradural tumors require detailed notification and follow-up for proper patient care.


Lymphangiomatosis is a benign proliferation of lymph vessels. Lymphatic diseases can vary from small lymphangioma to generalized lymphangiomatosis, which is a rare condition and can have several clinical manifestations. The gastrointestinal tract may be affected, but the incidence in the intestinal wall is very low. We propose in our study a case of ileal lymphangiomatosis presenting with perforation, in which the diagnosis was made after the pathological analysis of the resected intestinal tract. Although rare and not described in the literature, intestinal lymphangiomatosis could manifest itself with acute abdomen and could be a surgical urgency. This disease should be considered when intestinal perforation is observed. •Lymphangiomatosis is a multisystemic disorder.•The GI tract may be affected, but the incidence of lymphangioma of the intestinal wall is very low.•Lymphangiomatosis of the small bowel is a rare disease, so it is easy for a clinician to make a misdiagnosis.•Intestinal lymphangiomatosis could manifest itself with an acute abdomen and surgical urgency.•The pathologist should keep it in mind in the differential diagnosis, when he analyses a case of perforation. Lymphatic diseases can vary from small lymphangioma to generalized lymphangiomatosis, which is a rare condition and can have several clinical manifestations . AlthougWe propose in our study a case of ileal lymphangiomatosis presenting with perforation, in which the diagnosis was made after the pathological analysis of the resected intestinal tract. In addition, the relevant medical literature on intestinal lymphangiomatosis was reviewed. This work has been reported in line with the SCARE criteria .2A 41-year-old male presented to the Emergency Department with significant diffuse abdominal pain, nausea, vomiting and inability to pass gas or stool (constipation); the patient reported that these symptoms were present for 10 hours. He denied any significant family history of disease and he did not refer to major diseases or prior surgical interventions in his own medical history. During the physical examination, the subject was in discrete general conditions, collaborative, the sensory was intact, the decubitus indifferent, the breath eupneic and the pulse rhythmic. The vital parameters were preserved, and temperature was within normal limits. The abdominal examination revealed a flat abdomen, tender to palpation, painful to deep palpation on all quadrants, liver size appears within normal limits, Murphy's sign was negative, Blumberg's sign was positive, bowel sounds were absent. Rectal exploration indicated nothing significant. Laboratory values upon admission reported hemoglobin value of 15.5 g/dL, White Blood Cells 12.000/mmc, PCR 0.04 mg/dl. Upright abdominal films, then confirmed with computed tomography (CT) enhancement scan, revealed bowel distension and the presence of multiple gas-fluid levels. This framework suggested the presence of small bowel obstruction. As the etiology of the obstruction remained unidentified, the decision was made to perform a diagnostic laparoscopy. On entering the peritoneal cavity, the small bowel was examined: the intestinal loops were vital and vascularized. At approximately 80 cm from the ileocaecal valve, a volvulus was identified, and in close proximity there was a long tubular structure, which proved to be a Meckel's diverticulum, with adhesions to parietal peritoneum. A lysis of adhesions was performed, the Meckel's diverticulum was divided at the base using a linear stapler (45 mm) and a surgical drain was placed. On the first postoperative day, the patient showed an acute abdomen: untreatable-tensely distended abdomen, painful to deep palpation on all quadrant; Murphy's sign was negative, Blumberg's sign was positive; bowel sounds were absent. Surgical drain took out enteric material and blood. The body temperature was 38 °C. Laboratory values reported hemoglobin value of 12.7 g/dL, White Blood Cells 7.000/mmc, PCR 10.22 mg/dl. The patient again underwent urgent surgery. The intervention was an exploratory laparoscopy, then converted to open ileal resection for a microperforation of the bowel. The perforation site seemed to be distant above the previous diverticulum. About 20 cm of ileal resection was performed and side-to-side mechanical anastomosis was made. Two surgical drains were also placed. The postoperative course was regular, and the patient was discharged one the 10th post-operative day.Histological examination showed stratification of fibrin and granulocytes on the serosa and presence of diffuse lymphangiomatosis (positive immunohistochemistry of D2-40 marker) involving the submucosa and, in some parts, the full-thickness muscular wall , Fig. 2.3Lymphangiomatosis is a multisystemic disorder, characterized by congenital malformation of the lymphatic system with channels and cystic spaces of varying size. It may affect all the areas of the body; therefore, symptoms and complications are related to localization. Usually in the course of the disease patients are asymptomatic at first, but then the abnormally proliferating lymphatic channels are capable of massive expansion and infiltration into surrounding tissues. Abdominal lymphangiomatosis is quite often reported, but in many cases it arises in the mesentery, omentum, mesocolon and retroperitoneum. The GI tract may be affected, but the incidence of lymphangioma of the intestinal wall is very low, and it is even rarer in the small bowel (less than 1%) ,10. To tThere is only one case report of lymphangiomatosis involving the oesophagus  and one Lymphangiomatosis of small bowel is described in 6 works ,15,16. PIn the majority of patients described in the literature, diagnosis was suggested by colonoscopy and biopsy, which mainly showed protruding submucosal lesions. The diagnostic workup was sometimes integrated by contrast-enhanced CT, showing marked thickening of the walls of the bowel loops.In five papers ,15,16, plocus minoris resistentiae, and this may explain the pathogenesis of the perforation, reported in our case. The involvement can be continuous or, more frequently, segmental. The impairment of the muscular layer and of the submucosal nerve plexus could also have contributed to the development of the intestinal obstruction, which was the symptom because our patient came to our attention. This suggests that also lymphangiomatosis should be taken into account among other rare causes of intestinal obstruction [The histological features of the lymphangiomatosis are non-specific, and the definitive diagnosis requires the demonstration of an hyperproliferation of normal lymphatic vessels with normal endothelium, predominantly in the context of submucosa, with disruption of the muscular layer and sometimes of the serosa. This condition creates a truction ,20.4In conclusion, lymphangiomatosis of the small bowel is a rare disease that has no specific clinical features, so it is easy for a clinician to make a misdiagnosis or to miss diagnosis. In some cases, surgical resection may be required to provide definitive histological diagnosis, as occurred in our cases. We want to share our experience about this, because, although rare and not described in the literature, intestinal lymphangiomatosis could manifest itself with an acute abdomen and surgical urgency. This disease should be considered when intestinal perforation is observed. In particular, the pathologist should keep it in mind in the differential diagnosis, when he analyses a case of perforation whose cause is not very clear and specified.Ethical approval was not required.No fundingAntonio Giuliani, Lucia Romano: Writing.Gino Coletti, Mohammad Walid A Fatayer, Giuseppe Calvisi: Images and contribution to the text.Francesco Maffione, Chiara Muolo, Vincenzo Vicentini: Data collection.Mario Schietroma, Francesco Carlei: Study design and review.No conflict of interest.None.Prof. Francesco Carlei.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.Not commissioned, externally peer reviewed.


Hemangiomas are benign tumor formations of capillaries and blood vessels which are commonly found in various organs. However they are extremely rare in urinary bladder accounting for only 0.6% of all urinary bladder tumors. The differentiating of these benign tumors from malignant neoplasms are important since they have extremely different prognostic features as well as therapeutic strategies. Here we reported a female patient diagnosed with bladder cavernous hemangioma (BCH) after recent pelvic radiotherapy for the cervical cancer and reviewed relevant literatures.We reported a case of 49 years old female patient with persistent painless hematuria for 12 days. Computed tomography revealed a small lesion on the superior wall of the urinary bladder with acute clot retention. Cystoscopy confirmed a solid papillary pedunculated mass with a measuring of 1.0 × 0.5 cm located on the superior posterior wall and surrounded by distended vessels. Transurethral resection of the mass was then performed and the pathological report indicated a cavernous hemangioma of the urinary bladder. In a one and half year follow-up, no tumor recurrence or bleeding was found.The cavernous hemangioma in urinary bladder is rare and cystoscopic is a gold standard for diagnosis. Treatment options are vary for individuals and pathologic findings are vital for differentiating it from malignant potential tumors. A history of cancer related radiation therapy seems to be a risk factor for BCH. HoweverIt is important to carefully differentiate benign hematuria from malignant neoplasms such as angiosarcoma and Kaposi sarcoma, which are associated with immunosuppressed patients, since they have vital different prognostic features . Neverth2A 49 years old female was referred to a sudden, painless hematuria 12 days prior for her visit to our hospital. She had past history of cervical cancer about one year ago and received surgery as well as adjuvant chemotherapy and radiotherapy thereafter. The latest follow-up showed no evidence of tumor recurrence. Physical examination revealed no palpable mass or enlarged lymphnodes. She had also diagnosed with diabetes type 2 for 10 years with no medication pills treatment. Computed tomography revealed a small lesion on the superior wall of the urinary bladder with acute clot retention . CystoscWe then performed transurethral tumor resection and the tissue sample was sent to the pathological examination. Histological findings revealed the almina propria and submucosa of the urinary wall without infiltration of the muscularis propria A. They w3Most bladder tumors arise from the urothelium, and non-urothelial neoplasms are very rare in the bladder . These iHemangiomas are benign tumors formatted by capillaries and blood vessels, and the most common hemangiomas occurred in bladder is cavernous type, while much less frequent are capillary or arteriovenous types . Up to nUltrasonography is the first-line imaging method for the evaluation of gross hematuria, while other imaging tests such as computed tomography and magnetic resonance imaging are also useful in the diagnosis of vascular mass . The cysSince it is not commonly seen BCH in genitourinary tract, pathologists and doctors need to carefully differentiate it from malignant non-urothelial neoplasms, as they have extremely different prognostic features as well as therapeutic strategies ,14. The The therapeutic approaches for the management of BCH are vary in individuals due to their size, location and depth of penetration. Optional strategies includes observation, transurethral resection, electrocoagulation, radiation, systemic steroid administration, sclerosing agent injection, and partial cystectomy ,12. Part4The BCH is a benign non-urothelial tumor rarely occurred in the urinary bladder and treatment options are vary for individuals with favorable follow-ups. A history of cancer related radiation therapy seems to be a risk factor for BCH.None.National Natural Science Foundation of China (grant number 81700669), the Natural Science Foundation of Hubei Province of China (grant number 2016CFB217), and the China Scholarship Council.This work was supported by grants from This study was approved by Ethics Committee of Hubei Cancer Hospital.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.Xinming Hu – data collection, data analysis or interpretation, writing the paper.Kangli Deng – study concept or design, data collection, data analysis or interpretation, writing the paper.As this was a case report and not a clinical trial, this study does not require registration.Kangli Deng will be the corresponding author and take the full responsibility for the work and/or the conduct of the study, had access to the data and controlled the decision to publish.Not commissioned, externally peer reviewed.


Tumors of the ovary and appendix have been well documented in the setting of pseudomyxoma peritonei (PMP) with constant debate over tumor origin. Generally, these tumors are found to have a single primary origin, most commonly the appendix, with metastatic spread to the ovaries.Here we present a 61-year-old female who underwent total abdominal hysterectomy and bilateral salpingo-oophorectomy (TAH-BSO) for a primary mucinous ovarian carcinoma. She presented to our institution one year later with abdominal pain and a palpable right lower quadrant mass, which on histopathologic exam was found to be a primary low grade mucinous appendiceal neoplasm (LAMN), alluding to the potential of two separate primary disease processes.With two primary, non-synchronous lesions, a thorough literature review suggests that during the patient's initial TAH-BSO, she could have additionally undergone an appendectomy. In doing so, this would provide accurate, complete staging and determine if the two neoplasms were truly primary in origin or metastatic. In addition, new genetic markers are being discovered, such as the Special AT-rich sequence-binding protein 2 (SATB2) marker, which has been found to be positive in those with a LAMN and negative in those with a primary mucinous ovarian carcinoma. By acquiring appropriate and complete staging we can better diagnose and treat these neoplasms. Generally, these tumors are found to have a In this report, we discuss a patient who presented to the emergency room with right lower quadrant abdominal pain and a palpable mass for 2 months. Her past medical and surgical history included primary mucinous ovarian carcinoma with TAH-BSO and no chemotherapy/radiation. A computed tomography (CT) scan of the abdomen and pelvis demonstrated a 7.6 × 4 cm mass with peripheral calcifications suspicious for a mucocele versus malignancy. The patient underwent surgical exploration with excision of the mass that was well tolerated. Histopathology confirmed a primary low-grade appendiceal mucinous neoplasm.There are few documented cases of synchronous primary ovarian and appendiceal mucinous neoplasms; however, there have been very few recorded cases of two non-synchronous primary neoplasms and their appropriate diagnosis and treatment prompting this case report and literature review. Our work has been reported in line with the SCARE criteria .2Our patient is a 61-year-old Jamaican female who recently immigrated to the United States, with a past medical history significant for mucinous ovarian carcinoma status post TAH-BSO in Jamaica, 2018. The patient did not undergo any post-operative chemotherapy due to the low malignant potential of her initial pathology. Approximately one year later, she presented to our emergency department with right sided abdominal discomfort for the past two months. The rest of patient’s history was unremarkable. There were no other accompanying symptoms, namely change in bowel habits or weight loss. On physical exam, the patient had a palpable mass on the right side of her abdomen that was tender on palpation. A CT scan of the abdomen and pelvis demonstrated a 7.6 × 4 cm mass with peripheral calcification suspicious for a mucocele versus mucoid epidermoid carcinoma . SubsequWith a presumed diagnosis of appendiceal mucocele versus malignancy, the patient agreed to surgical intervention. The patient was taken to the operating room for an exploratory laparotomy. Upon inspection of the abdominal cavity, the patient was found to have a mass in the right lower quadrant which appeared to be originating from the appendix. Numerous omental implants were identified warranting an omentectomy, as well as several deep pelvic gelatinous deposits which were removed with sharp dissection. The right lower quadrant mass appeared intact, freely mobile, and limited to the distal tip of the appendix. There was limited surrounding inflammation, with mild fibrous adherence to the right paracolic gutter. After careful dissection, the appendix with the associated distal mass were removed and sent for pathology.2.1The omentum was found to have a focus of low-grade mucinous neoplasm consistent with an appendiceal origin. The appendix was found to be a low grade appendiceal mucinous neoplasm with peritoneal involvement . The proThe patient had an uneventful postoperative course and recovered without complications. She was discharged on postoperative day three with continued follow up in our clinic.3Although a distended, mucus-filled appendix is often called a mucocele, this term is ambiguous and best used to describe a radiological finding rather than a pathologic entity. In 2012, the Peritoneal Surface Oncology Group International (PSOGI) developed a consensus classification that has helped to resolve much of the confusion surrounding diagnostic terminology [These lesions are rare, with only one to two thousand cases diagnosed annually in the United States . WhetherThe LAMN is defined as a true neoplasm with abundant mucin production as well as dysplastic epithelium. Despite their bland appearance, LAMNs may penetrate through the appendiceal wall, cause appendiceal rupture, and progress to pseudomyxoma peritonei (PMP) . StagingThere has long been debate of which tumor, ovarian or appendiceal, is the site of origin in the setting of pseudomyxoma peritonei (PMP). PMP is a rare disease in which the mucin cells are distributed within the peritoneal cavity. Although most literature states PMP’s origin is appendiceal in nature, many other tumors including ovarian, stomach, and pancreas have been documented. These mucinous cells continue to proliferate which eventually causes the signs and symptoms seen during patient presentation. The tumors continue to grow and can perforate, causing seeding with the peritoneal cavity and leading to possible metastasis.This patient was found to have a primary mucinous ovarian carcinoma in early 2018 diagnosed in her home country of Jamaica. In March of 2019, once she immigrated to the United States, she was found to have a primary mucinous appendiceal neoplasm (LAMN) with cyto-histopathology confirming no primary ovarian lesion. In the available literature, synchronous tumors of the ovary and appendix are an uncommon yet well-recognized occurrence in the setting of PMP [In 2018, our patient was diagnosed with the mucinous ovarian carcinoma and underwent a (TAH-BSO) with no further treatment thereafter. A recent 2017 study conducted in Denmark, published in the International Journal of Gynecological Cancer, aimed to assess the importance of an appendectomy in the presence of a mucinous ovarian adenocarcinoma as it can be difficult to distinguish between primary ovarian and primary appendiceal cancers clinically, histologically, and immunohistochemically . EssentiThere is an extensive immunohistochemical panel that assesses expressions of various markers; however, even then, differentiation is difficult and pathologist dependent. Our patient’s current immunohistochemical analysis tested for CK7 and CK20 which help differentiate an appendiceal versus ovarian mucinous neoplasm. With recent advancements in gene sequencing, the March 2018 issue of Pathology Research and Practice published an article with evidence of a new supportive marker for the differentiation of a primary mucinous tumor of the ovary and an ovarian metastasis of a low-grade appendiceal mucinous neoplasm (LAMN), the Special AT-rich sequence-binding protein 2 (SATB2) marker. The SATB2 expression is primarily within the gastrointestinal tract. Patients with physical, radiographic and/or pathologic evidence of an ovarian mass with concomitant LAMN, in the context of pseudomyxoma peritonei (PMP) or small foci of peritoneal spread, the addition of the SATB2 marker in immunohistochemical staining revealed strong expression of the marker in those with LAMN while SATB2 was negative in cases that demonstrated cyto-histopathologic primary mucinous ovarian carcinomas . Our pat4The presented case demonstrates a rare variant in mucinous neoplasms: a primary ovarian and primary appendiceal neoplasm diagnosed and treated approximately one year apart in two different countries. Although there is no literature to support a genetic anomaly that increases the risk of mucinous tumors throughout the body, with gene sequencing and newly developing biotechnology, there may be a biologic component not currently recognized. In addition, diagnosing mucinous neoplasms based on pathology is challenging even amongst seasoned pathologists. More detailed immunological studies with more than one pathologist overlooking the case may be warranted as new markers, including the SATB2, continue to be discovered. Furthermore, adequate staging is a necessity because of this disease diagnostic challenges and routine appendectomy in the setting of primary ovarian mucinous neoplasms may be warranted to achieve the proper diagnosis and treatment for patients. These new markers and staging protocols can continue to help us accurately diagnose primary versus metastatic disease processes and achieve the best outcomes for our patients in the future.In our case, with stage T4a LAMN, regularly scheduled imaging and tumor marker follow up can permit earlier detection of recurrent disease, which occurs between 4.8 and 20.4 % of the time . CurrentThere were no study sponsors involved in the submitted work.This is not applicable to the submitted work.Written informed consent was obtained from the patient for publication of this case report and accompanying images.Study Concept/Design – Misbah Yehya, Matthew Denson, Zbigniew Moszczynski.Data Collection - Misbah Yehya, Matthew Denson, Zbigniew Moszczynski.Writing the Paper – Misbah Yehya.This is not applicable to our submitted work.The Guarantor’s are Misbah Yehya, Matthew Denson, and Zbigniew Moszczynski.Not commissioned, externally peer-reviewed.There are no conflicts of interest that influence the submitted work.


Anatomical variants of the extrahepatic biliary tree are numerous, adding significantly to the risk of bile duct injury during cholecystectomy, especially when laparoscopic approach is employed. Duplicated cystic ducts draining a single gallbladder are extremely rare.A 34-year-old female presented with signs and symptoms of acute cholecystitis which was confirmed on imaging. She was found to have an accessory cystic duct on laparoscopic cholecystectomy requiring conversion to open laparotomy with intraoperative cholangiogram to delineate the anatomy.In the English literature, there has been 20 reported cases of double cystic duct with a single gallbladder. Most of these cases were diagnosed intraoperatively despite the completion of a preoperative endoscopic retrograde cholangiopancreatography in a few of these patients.The limited success of preoperative biliary tract imaging in demonstrating anatomic aberrancies prior to cholecystectomy clearly highlights the importance of maintaining constant vigilance for even the slightest anatomic abnormality at operation. Any uncertainty or concern for ductal injury mandates immediate operative cholangiogram with cannulation of all structures in question. The bas2We report a case of a 34-year-old female who presented to the emergency department with three days of constant right upper quadrant and epigastric abdominal pain with associated nausea. The patient had similar pain two months ago that resolved and did not seek medical attention. On presentation she had normal vital signs. Physical examination demonstrated right upper quadrant tenderness without peritoneal signs with negative clinical Murphy’s sign. Her blood work revealed a white blood cell count of 13,500 /microliter, alanine aminotransferase of 318 U/L, aspartate aminotransferase of 259 U/L, alkaline phosphatase of 120 U/L, and total bilirubin of 1.7 mg/dL. Ultrasonography demonstrated new mild gallbladder wall thickening and negative sonographic Murphy’s sign with equivocal suggestions of acute cholecystitis. Hepatobiliary iminodiacetic acid scan was subsequently performed and showed non-visualization of the gallbladder consistent with acute cholecystitis.The patient was taken to the operating room and the cystic duct and artery were dissected free from the cystic triangle laparoscopically. Both structures were secured proximally and distally and divided sharply. The gallbladder was dissected from the bed using electrocautery. Due to the contracted and intrahepatic nature of the gallbladder, approach was switched to a top-down technique. Just prior to removal of the gallbladder from the liver bed, another structure entering the gallbladder was encountered . At thisPostoperatively, the patient’s liver function tests normalized. Patient was discharged home on postoperative day 3 and was tolerating diet. She was seen in the surgical clinic 2 weeks postoperatively and was doing well. The pathology report showed acute on chronic cholecystitis with mucosal ulceration and cholelithiasis.3Intrigue with anomalous gallbladder anatomy and associated extrahepatic biliary duct aberrancy originates as far back as 1926 with Edward Boyden’s comparative report and classification of the various congenital anomalies of the gallbladder . Thirty In the English literature, there has been 20 reported cases of duplicate cystic ducts draining a single gallbladder including our reported case  Double cystic duct was identified intraoperatively in 16 out of the 19 patients (84%) who were operated on. Despite the completion of a preoperative endoscopic retrograde cholangiopancreatography (ERCP) in 7 patients, the cystic duct anomaly was only identified in 3 cases (43%) ,12,20. T4The limited success of preoperative biliary tract imaging in demonstrating anatomic aberrancies prior to cholecystectomy clearly highlights the importance of maintaining constant vigilance for even the slightest anatomic abnormality at operation. Any uncertainty or concern for ductal injury mandates immediate operative cholangiogram with cannulation of all structures in question. Although laparoscopic cholecystectomy is safe in experienced hands, a low threshold for conversion to laparotomy should be had when the anatomy cannot be deciphered.No conflicts of interest to be declared.No source to be stated.The study is exempt from ethical approval in our institution.Written informed consent was obtained 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.Semeret Munie: Formal analysis; Writing – original draft.Hassan Nasser: Writing – review & editing.Pauline H. Go, MD: Writing – original draft.Kelly Rosso, MD: Visualization.Ann Woodward: Supervision.Not applicable.Ann Woodward, MD.Not commissioned, externally peer reviewed.


Acute portal and superior mesenteric vein thrombosis(SMV) is a rare but potentially lethal condition that is often characterized by generalized and non-specific symptoms. A high index of suspicion is warranted for early diagnosis and management. We present a case of 54 year old male who presented with generalized abdominal pain which was later accompanied by hemodynamic instability and radiological diagnosis of portal vein and superior mesenteric vein thrombosis.The management of SMV can be divided into medical and surgical therapy. Patients who have clear signs of peritonitis will require emergent surgery. Interventions for life-threatening portal vein thrombosis are limited and poorly described in the literature.We report a case of extensive portal vein thrombosis(PVT) advancing proximally to involve the superior mesenteric vein. Rapid portal vein patency and improved hepatic function was achieved with the direct use of tissue plasminogen activator infusion via operatively placed middle colic vein catheter. PV and SMV thrombosis may occur as a consequence of medical conditions such as cirrhosis, pancreatitis or surgical interventions such as a splenectomy . PVT mayWe present a case of PVT and mesenteric vein thrombosis and attempted management with direct intraoperative middle colic vein thrombolytic infusion.The following case report has been reported in line with SCARE criteria  which satisfies one of the criteria suggestive of autoimmune pancreatitis [Although systemic anticoagulation with heparin is the traditional therapy for acute portal and superior mesenteric vein thrombosis, catheter directed thrombolysis may be a resourceful adjunct in the management of this potentially lethal condition. Early Surgical Intervention with resection of infarcted bowel followed by post-operative anticoagulant therapy is crucial for promising outcomes.The following is a disclosure that the following case report holds no financial or otherwise personal relationship with other people or organizations that may influence our work. No conflict of interest is present.The following manuscript did not involve any funding resource for any part of the process.Research approval has been attained by the hospital body ethics committee, furthermore consent for writing up the case report while maintaining anonymity was taken from the patient.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is avaialble for review by the Editor-In-Chief of this journal on request.Writing the paper: Dr. Maha Marshad.Data Collection: Dr. Maha Marshad.Data Analysis: Dr. Maha Marshad, Dr. Thamer Al Abbasi.Literature Research: Dr. Maha Marshad.Manuscript Editing and Review: Dr. Thamer Al Abbasi, Dr. Martin Maresch.Manuscript Finalization: Dr. Maha Marshad, Dr. Thamer Al Abbasi.N/A.Dr. Maha Marshad.Commissioned, externally peer-reviewed.


This report illustrates a case of asymptomatic bilateral orbital varices in a 64-year-old Caucasian male. The orbital varices were incidentally discovered while investigating the patient’s initial presentation of bilateral chorioretinal scars and optic nerve head drusen. Magnetic resonance imaging (MRI) of the brain and orbits with contrast confirmed the presence of bilateral varices of the inferior ophthalmic veins and pterygoid plexuses. The occurrence of bilateral orbital varices is quite rare, and few asymptomatic cases have been described in the literature. Orbital varices typically result from a congenital weakness in the postcapillary venous wall . This caA 64-year-old Caucasian male with a medical history of type 2 diabetes mellitus, hypertension, and hyperlipidemia presented to an eye clinic for a diabetic eye exam. He had no ocular complaints aside from slightly blurred vision, which he attributed to “scarring on his retina.” He stated that he had developed “smoky vision” several years ago, which had been treated with oral and topical medications. He denied any current ocular discomfort or pain.+2 in the right eye and 20/20-2 in the left with correction. Anterior segment examination did not reveal any abnormalities. Intraocular pressures were within normal limits. Dilated fundus examination found optic nerve head drusen, extramacular healed chorioretinal scars, and mild non-proliferative diabetic retinopathy in both eyes.On exam, his visual acuity was 20/25Diagnostic work-up included laboratory testing and magnetic resonance imaging (MRI) of the brain and orbits with and without contrast. This work-up was prompted by the history of chorioretinal scarring and blurry vision in the past requiring treatment. Laboratory workup was negative aside from positive toxoplasma IgG antibodies. MRI of the brain and orbits with and without contrast revealed varices of the bilateral inferior ophthalmic veins, bilateral pterygoid plexuses, and the infratemporal veins. There was no dilation or thrombosis of the superior ophthalmic veins, and no abnormal enhancing mass lesions within the orbits or brain parenchyma. No intracranial arteriovenous malformations, dural fistulas, or carotid cavernous fistulas were identified Figures -3.A venous varix is a dilation of one or more veins associated with low blood flow and distensible walls. Orbital varices are a rare condition, accounting for less than 1.3% of all orbital masses . AlthougThe orbital venous system is highly variable compared to other venous systems elsewhere in the body. The inferior ophthalmic venous plexus has multiple valveless interconnected veins with extremely small diameters and short lengths . The orbOrbital varices can be divided into primary and secondary orbital varices. Primary orbital varices are idiopathic and confined to the orbit. Secondary orbital varices are acquired, and usually a result of a carotid-cavernous fistula, dural arteriovenous fistulas, or intracranial arteriovenous malformations -4.Primary orbital vascular malformations are believed to be present at birth. Even though it is thought to be congenital, patients do not typically become symptomatic until they are young adults. However, cases have been reported across all age groups -4. TheseIn addition to the typical presentations, many patients will be diagnosed due to a sudden complication. Orbital venous varices are the most common cause of spontaneous intraorbital hemorrhage . Acute tImaging modalities to detect varices include ultrasound, color Doppler imaging, computerized tomography (CT), and MRI including magnetic resonance venography (MRV). During imaging, dynamic maneuvers such as Valsalva, in conjunction with specific positioning, can aid in the visualization of varix . OrbitalFor asymptomatic varices, observation with serial imaging is the usual method of follow-up. This reported patient will be monitored and imaged periodically. For symptomatic varices causing a mass effect, disfigurement, vision loss, optic nerve compression, thrombosis, or hemorrhage, surgical intervention is warranted with a multidisciplinary approach. Generally, orbitotomy with excision of the varices is the treatment of choice. Varices are easier to excise with less invasive modalities if the anterior portion is thrombosed, with identification being more difficult in a supine patient with non-thrombosed varix -4. RecurThere can be significant variations in the vascular pattern of orbits between individuals. This case report illustrates the extremely rare combination of bilateral orbital varices in a completely asymptomatic individual.


It is well known that cavernous malformations become much more hemorrhagic after the initial hemorrhage and that brainstem lesions are more dangerous than the lesions in the supratentorial location. It is very difficult to handle symptomatic cavernous malformations associated with repetitive hemorrhages in the brainstem. Patients may be suffering from brainstem syndromes such as hemiparesis, hemisensory disturbance, ataxia, and disturbed ocular movement. We have encountered two such difficult cases, one is very large and the other is multiple and familiar, accompanying repetitive brainstem hemorrhages. Since microsurgery seems to be very difficult and hazardous, these two cases were treated with radiosurgery after several hemorrhages. Intracranial cavernous malformations (CMs) are congenital vascular anomalies similar to arteriovenous malformations, venous malformation, and capillary telangiectasia . They arCase 1A 26-year-old male presented with no previous medical or surgical history. His illness started with occasional head heaviness over the right retromastoid portion. He complained of no weakness of the arm or leg, but sometimes complained of mild weakness of his left hand. He felt some difficulty in closing his eyes. After the initial complaint, he experienced headache several times and felt mild weakness and dysesthesia in his left arm and leg. When he experienced dysesthesia several times, he had to stop moving. No convulsive seizure occurred.The first CT scan Figure  showed aThe core of CM encircled with a yellow line, not including the surrounding low-intensity rim, measuring 1 mL in volume was treated by Gamma Knife radiosurgery with a marginal dose of 12 Gy (Figure He experienced a fluctuating dysesthesia on the left half body for a while after the treatment, but no apparent weakness of the arm and leg occurred during nine months after Gamma Knife treatment. MRI showed no obvious changes in the CM, but showed a minor decrease in mass effects Figure . He retuCase 2A 41-year-old woman presented to our hospital seeking preventive methods of further bleeding subsequently after the last bleeding episode in the midbrain. She revealed that she had a sudden unconsciousness attack associated with nausea and vomiting at the age of 12 years. She was diagnosed as having brainstem hemorrhage due to CM, and multiple CM lesions in the entire brain. Her disturbed consciousness recovered within several months. She had an apparent hereditary background, and many of her relatives had been suffering from the same or similar disorders of the central nervous system (Figure Thereafter, she had shown full recovery and had been well for the following 16 years. However, at the age of 28 years, she developed a second hemorrhage in the pons and a subsequent episode in the medulla. By the third attack and sudden onset of left hemiparesis and ophthalmoplegia, she was unable to walk. At this time, CT and MRI showed isolated hematoma in the left peduncle of midbrain. Several small CMs were confirmed in the supratentorial portion, which were not associated with an apparent hemorrhage Figure .Since the CMs were in the pons, medulla, and midbrain, radiosurgery was performed one by one in two sessions, with the marginal doses of 11 Gy. After the radiosurgery, she has been well except for complex partial seizures associated with motor arrest and transient faintness, which occurred once or twice in a month. Follow-up MRIs after 12 months showed no further hemorrhage, and the lesions in the brainstem became smaller in size Figure . She hasThere has been a certain agreement that CMs rarely cause bleeding or seizure. Therefore, conservative treatment has been chosen for incidentally found CMs since the majority of them are believed to be asymptomatic overtime. Curling et al.  estimateSurgical resection of s-CMs is apparently preferable to prevent further hemorrhages and intractable seizures. However, surgery is not always safe because CMs may originate in eloquent locations such as the brainstem or basal ganglia. Therefore, a selection bias for surgery is apparent, and only superficial as well as easily accessible CM lesions are currently operated. Nonetheless, several reports on surgical resection -9 are ofRadiosurgery with a Gamma Knife has been used in s-CMs in the brainstem as a treatment option. In the literature -15, succTwo difficult cases of s-CM in the brainstem are reported, one is large and the other is multiple and familial. Both cases were not eligible candidates for microsurgery, and radiosurgery seemed to be a choice. It is crucial to shape up the target volume for large lesions, and only the core of CM should be treated with an adequate marginal dose of less than 13 Gy. On the contrary, it is important to select the dose and the timing either with a single session or multiple sessions when the lesions are multiple and adjacent to each other.


We report the case of a 79-year-old male who presented with altered bowel habit. Subsequent endoscopy and radiological investigations suggested the diagnosis of a likely sigmoid colon malignancy. Surgery was performed to remove the suspected tumour and post-surgical histology showed only inflammatory change in an area of underlying sigmoid diverticulosis with an associated impacted dental prosthesis. Occasionally, these pass through to the stomach and into the small and large bowel where they can either pass through without incident or become impacted and cause an obstruction and/or perforation.2Within the elderly population the aspiration/ingestion of dental prosthesis is more common.4 The base material used to make most removable dental prostheses, polymethylmethacrylate (PMMA), is radiolucent;6 this creates a diagnostic challenge following accidental ingestion as radiological tracking of the ingested denture/partial denture becomes difficult.Plain radiographs are the mainstay initial investigation for swallowed foreign bodies, especially dental material, as most dental prostheses are radiopaque.We present a case where impaction of the partial denture within the sigmoid colon mimicked carcinoma on endoscopy and subsequently on radiological evaluation.A 79-year-old male was referred to the surgical outpatient department with a 2-month history of altered bowel habit. Abdominal examination and digital rectal examination in the outpatient clinic were unremarkable.The laboratory studies, full blood count and biochemistry were normal. A colonoscopy study identified four abnormal lesions, two of which were colonic subcentimetre polyps. More distally in the mid-rectum, at 15 cm from the anal verge, there was a sizeable, villous polypoidal lesion, which was proved on biopsy to be a tubulovillous adenoma (TVA) showing low-grade dysplasia. More proximally in the distal sigmoid colon at 25 cm there was a segment of a malignant-looking lesion associated with a stricture . HoweverAs per local protocol for malignant-appearing lesions on endoscopy, a staging CT scan was arranged on the basis of the colonoscopy appearances to facilitate the cancer pathway. This demonstrated, at the site of the presumed malignancy in the distal sigmoid colon, a 7 cm segment of circumferential thickening on a background of diverticulosis . This waAlthough the 5 cm rectal TVA could explain the patient’s symptoms it was felt at the multidisciplinary team discussion that either lesion could be responsible for the patient’s symptoms. Despite the lack of malignant cells from the biopsy samples the surgical team were still concerned about a potential sigmoid colon malignancy from the endoscopy and CT appearances. It was felt that the colonoscopy sampling was not truly representative of the malignant appearing sigmoid colon lesion seen at colonoscopy and CT. An anterior resection was planned for the patient to remove both the TVA and presumed sigmoid colon malignancy. An MRI scan of the rectum was performed in order to preoperatively assess the rectal polyp further. The sigmoid tumour was incidentally included in the MRI scan field owing to proximity to the rectal lesion ; howeverThe patient underwent an uncomplicated laparoscopic-assisted anterior resection with defunctioning loop colostomy. Post-surgical histology confirmed that the rectal polyp was a low-grade TVA. However, histological assessment of the presumed distal sigmoid cancer at 25 cm demonstrated mucosal ulceration with underlying fibrosis and diverticulosis. The ulceration was associated with a 5 cm jagged edged intraluminal dental plate containing a single front upper incisor prosthetic tooth found impacted at this site, with no metallic components . The denRetrospective review of the patient’s previous imaging revealed that the patient had attended the accident and emergency department 2 years earlier, having accidentally swallowed “false teeth” during a meal. An abdominal plain radiograph had been taken at the time, which did not demonstrate any radiopaque foreign body. The CT scout view also demonstrates no radiopaque foreign body . The pat7 However, as in the case patient, more distal colonic impaction of foreign bodies may result in serious late complications, especially in the presence of significant colonic diverticulosis. The prevalence of colonic diverticulosis is 65% from the age of 80 years onwards8 and risk of aspiration is also high in this age group. There is a higher risk of impaction associated with dental prostheses owing to their size.Most available literature on cases of complications of accidentally swallowed dental prostheses describes acute oesophageal impaction and related complications owing to the size of the prostheses.4 failure to pass through the alimentary canal after several weeks should also raise the possibility of impaction—particularly at an area of narrowing such as the ileocaecal valve or a colonic lesion causing luminal narrowing such as an area of significant diverticular change. In the case reported by Tanrikulu et al,9 the swallowed lower denture was radiopaque and surgery was performed when ileocaecal impaction occurred leading to obstructive symptoms. In another case reported by Ghori et al,1 sigmoid perforation from the partial denture occurred with a fatal outcome to a young patient despite surgical management.Just as failure of migration on imaging is concerning for obstruction or perforation for swallowed radiopaque materials,In this case, it is not entirely clear which lesion would have caused the patient’s symptoms but surgery was necessary in order to alleviate his symptoms. Given the location of the dental prosthesis and the duration of 2 years since ingestion it is likely that the relatively recent bowel symptoms were all owing to a more recent lesion such as the TVA. However, with progression of the sigmoid diverticulosis and associated inflammatory changes future complications would still be possible had surgery not been performed.The question remains whether all dental prosthesis should have a radiopaque marker to assist with tracking in the event of accidental ingestion/aspiration. Retrospective analysis of the contrast-enhanced CT scan images reveal a thin linear area of slight hyperdensity in the intraluminal region of the sigmoid colon lesion  that cou7 Although this is similar to the CT findings in our case patient,  for publication of this case report, including accompanying images.


Solitary fibrous tumours (SFTs) rarely occur in the orbit, especially in the lacrimal area. These tumours are mostly solid. Cystic changes have been documented, but they remain very rare. Only three cases of primary orbital solitary fibrous tumours with cystic changes have been reported in the literature, but no cases have been reported to occur in the lacrimal gland. Solitary fibrous tumours generally follow a benign course and are treated definitively with surgical excision. Data from the literature suggest that the cystic nature of SFT presents a risk of recurrence and could be a harbinger of malignancy.A 42-year-old woman was admitted to the endocrinology department for right unilateral exophthalmia and epiphora in the last 8 months. An ophthalmological evaluation showed exophthalmia only in the right eye (22 mm) and normal visual acuity, visual field and extraocular movements. Investigations revealed normal thyroid function. Orbital magnetic resonance imaging detected a 4 × 2,2 × 2,7 cm septate pseudocystic mass in the right lacrimal gland. Given her lacrimal gland tumour diagnosis, the patient was submitted for neurosurgical intervention with total ablation of the tumoural mass and complete right dacryoadenectomy. Although the intraoperative extemporaneous examination results were suggestive of a haemangiopericytoma, histological and immunocytochemical examination showed an extrapleural SFT. The postoperative clinical evolution was favourable, with remission of the exophthalmia. Fifteen months after surgery, no signs of recurrence were noticed.We report the first case of an SFT with cystic changes in the lacrimal gland. Although the presence of cavitary lesions alone does not necessarily indicate aggressive behaviour, cystic changes pose a risk of recurrence and may suggest malignant transformation over time. As a result, our case requires long-term follow-up due to recurrence and malignant potential. Although until a few years ago the most frequent site of solitary fibrous tumours (SFTs) was thought to be the intrathoracic region, with a peculiar predilection for body cavity sites, this tumour can develop in any part of the body where mesenchymal cells are present. Orbital involvement was first independently reported in 1994 by Dorfman et al. and Westra et al., respectively . Until nWe introduce a case of lacrimal SFT with cystic changes and a brief literature review regarding this specific topic. This research is based upon the collection of data from all cases of SFT involving the lacrimal gland area reported in the English-written literature starting with the first case reported in 1996 and including our current case.We present the case of a 42-year-old Caucasian non-smoking female who was admitted in the Endocrinology Department for right unilateral exophthalmia and epiphora in the prior 8 months Fig. a. Her faIn adolescence, the patient was diagnosed with allergic rhinitis, for which she received inconstant Xyzal. There was no inflammatory disease or previous trauma in the anamnesis.2 and her clinical data were normal. The ophthalmological evaluation showed only exophthalmia of the right eye (22 mm) and normal visual acuity, visual field and extraocular movements. No optic nerve atrophy or papilledema was found. The neurological exam was normal. No abnormal findings were identified on current laboratory tests, including a full blood count, erythrocyte sedimentation rate, glycaemia, renal profile, and liver function. The hormonal thyroid profile was within the normal range and showed no autoimmune abnormality. Ultrasonography of the anterior cervical region showed a thyroid with normal volume and structure. Under these circumstances, an orbital magnetic resonance was recommended. A well-defined 4 × 2,2 × 2,7 cm mass in the right lacrimal gland with septate pseudocystic appearance, small haemorrhagic areas, heterogeneous structure, and mass effect on adjacent structures without local bony remodelling was detected  was 24,8 kg/mted Fig. b, c. TheBased on this diagnosis, the patient was referred to a neurosurgical department. The lesion was approached extraperiosteally through a sub-brow incision. The tumoural mass with swollen lacrimal gland was identified in a clear dissecting plane and isolated from the surrounding muscles and showed no local infiltration Fig. a. Total Macroscopically, the tumour specimen was well-circumscribed and had a bosselated surface, a purple greyish colour, yellow areas and an elastic consistency and measured 2,5 × 1,7 × 1,5 cm in size Fig. a. The diThe postoperative clinical evolution was favourable, including remission of the exophthalmia Fig. d but perSFTs represent a diverse group of ubiquitous rare spindle cell neoplasms with variable clinical behaviour and have not yet been fully explored. SFT was first described in 1931 by Klemperer and Rabin, who reported it as a primary neoplasm of the visceral pleura. Although once considered to originate from mesothelial cells, with the advent of immunohistochemistry, a fibroblastic origin, occasionally with myofibroblastic differentiation, is now accepted . AdditioAll patients with lacrimal SFTs present one or more symptoms relating to a growing tumoural mass at the site of the lacrimal gland. This may be accompanied by upper eyelid swelling, occasionally limited ocular motility, unilateral eyeball displacement and facial deformity. In our case, there was no palpable mass. Sometimes, localization of the tumour in the lacrimal area causes eyeball protrusion, leading to misinterpretation as endocrine exophthalmia and the referral of the patient to an endocrinology service, as occurred in our case. These tumours have previously tended to grow slowly, form a single mass, and sometimes exert compressive effects on the adjacent viscera or neurovascular structures; the exception was one case that did not exceed 3.5 cm in size.In addition to a complete physical examination, affected patients should undergo imaging tests that may include X-rays, ultrasound, computed tomography scan or magnetic resonance imaging (MRI). In the previously reported cases, a well-demarcated mass with regular margins and no infiltrating adjacent structures or bone remodelling was described. MRI features can be variable, are correlated with cellular components and collagen, degenerative changes, haemorrhage and necrosis within the tumours. Compared with the cerebral cortex or extraocular muscle, SFTs in the lacrimal area show isointense signals on T1-weighted images and variable signals on T2-weighted images . In addiBecause of variable clinical presentations and histologic appearances, the diagnosis of SFTs can be a challenge, as we observed in our case, when the result of an extemporaneous examination is not confirmed by the results obtained in paraffin-embedded tissues and immunohistochemistry. SFTs can be misdiagnosed as other mesenchymal lesions , smooth muscle tumours (leiomyomas and leiomyosarcomas), neural tumours , etc. In the past, some SFTs have probably been misdiagnosed as other spindle cell neoplasms. An extensive histopathological analysis of 41 fibroblastic lesions of the orbit that had different diagnoses, including haemangiopericytomas, fibrous histiocytoma, mixed tumours and giant cell angiofibroma, led to their reclassification as SFTs .Histopathological and immunohistochemical staining features and, more recently, molecular analyses are the basic tools used to diagnose SFTs and also have prognostic value. Additionally, fine needle aspiration has been mentioned as useful for diagnosing SFT . SimilarImmunohistochemical tests are necessary to confirm the diagnosis. CD34 has over time become considered the most sensitive marker for SFTs (95–100%); therefore, to call a tumour an ‘SFT’, it should be CD34-positive . In all Cytogenetic studies are also needed to improve understanding of the clinical and pathological features of SFTs. Although research is underway, some karyotypic aberrations, including both numerical and structural abnormalities, have been reported , 21. AddRecent molecular analyses suggest that SFTs are positive for nuclear protein STAT6, which is overexpressed due to a paracentric inversion on chromosome 12q13 and NAB2-STAT6 gene fusion , 24. TodAnother gene that is upregulated in SFTs is GRIA2, which encodes a subunit of a class of glutamate receptors thought to mediate increased cell proliferation. In a recent study, this marker, which has not yet been researched in lacrimal gland SFTs, was found to be positive in 89% of SFTs and negative in more than 99% of all other histological mimics occurring in soft tissue . HoweverAs in other sites of SFTs, effective treatment of SFT of the lacrimal gland and lacrimal fossa means complete resection of the tumour. Sometimes, the entire lacrimal gland may have to be removed, as occurred in our case. No other treatments (radiotherapy or chemotherapy) have proved to be efficient. According to some authors, preoperative arterial embolization can be considered because of the risk of bleeding during surgical resection of these highly vascularized tumours .A correct diagnosis is mandatory for proper therapy and management of patients with SFT. Until now, it has been estimated that most extrapleural tumours, with the exception of those of mediastinal origin, have a benign outcome. Usually, recurrences have been attributed, for the most part, to incomplete resection. A review of 189 cases of CNS SFTs revealed that the rate of recurrence in incomplete resection was 58,1%, whereas the rate was 14% in cases of total excision . In addiIt is unclear whether SFTs occurring in the lacrimal gland exhibit recurrences or malignant behaviour. To date, no cases with recurrence or metastasis have been reported, suggesting a possible non-aggressive evolution for lacrimal gland SFTs. However, these results should be interpreted with caution because statistical analyses were performed on only a small number of reported cases. Could the recurrence or malignant behaviour of these tumours be correlated with their morphological features? Although orbital SFTs are mostly solid tumours, partial or complete cystic changes have been reported in seven previous cases (three primary and four recurrent), but none of these occurred in the lacrimal gland. –36. AlthWith new advances in medical research, more cases of orbital solitary fibrous tumours, including SFTs in the lacrimal area, are being diagnosed and reported. To date, in all previously reported cases, SFTs of the lacrimal gland were solid tumours, and cystic changes were not been described prior to our case. Cystic degeneration of the tumour requires long-term follow-up because recurrence at the site of surgery or metastasis can occur several years after the excision of the primary tumour, even in tumours that behave benignly.


Invasive moles occur in the fertile period, with about 95% occurring after previous mole removal and the remaining 5% occurring after several other pregnancies.A 27-year-old patient developed a rare invasive mole two months after a missed abortion.A transvaginal ultrasound scan revealed a 3.6 × 2.9 × 2.4 cm sized lesion with cystic vascular areas within it, within the myometrium of the right fundal posterior region of the uterus. There was no metastasis to other organs.TM.After administration of methotrexate, the level of beta-human chorionic gonadotropin (ß-hCG) was elevated and liver enzymes were also markedly elevated. She wanted to retain fertility for future pregnancies. After laparoscopic removal of the myometrial invasive mole, the incision site was sutured with a 3-0 V-LocOne year later, a natural pregnancy occurred and a cesarean section was performed at 36 weeks.This is the first reported case of its type. Our case demonstrated that pelviscopic removal of an invasive mole is possible if there are no other metastases, and that future pregnancy and childbirth are still feasible in women of reproductive age. Ultrasonography corresponds with the classical clinical presentation and an accurate diagnosis is possible in a majority of cases. With early and accurate diagnosis, the cure rate is high.Invasive H-moles or invasive moles are a type of abnormal pregnancy that grows into the muscular wall of the uterus. It may aggressively invade the myometrium and may even perforate the uterine serosa. The most common life-threatening complication of invasive moles is uterine perforation. After rupture of the uterus, massive bleeding, hemoperitoneum, and acute abdominal pain occurs. 2TM absorbable wound closure device   levels. Her physical examination was unremarkable. Her gynecological history was notable for the occurrence of an abortion at 12 weeks gestation six months previously, which turned out to be a missed abortion. She subsequently underwent two suction and curettage procedures, as the ß-hCG was persistently elevated to 50,063 mIU/ml. Following this, however, the ß-hCG still continued to rise, reaching 17,008 mIU/ml. The patient underwent two more dilatation and curettage procedures and was given an injection of 50 mg methotrexate around one month prior to presentation at local clinic. Menstruation had still not returned. Serial ß-hCG tests were performed, and levels initially fell to 68 mIU/ml, then remained constant around 60 mIU/ ml for eight weeks, at which time she was referred to our hospital. A transvaginal ultrasound scan revealed that both ovaries were unremarkable. Endometrial thickness was 2 mm. A 3.6 × 2.9 × 2.4 cm sized heterogenous hyperechoic solid lesion with cystic vascular areas within it, was noted within the myometrium of the right fundal posterior region of the uterus, away from the endometrium. The thickness of the remaining muscle layer with invading mole was very thin, 2 mm  has established the following guidelines for the diagnosis of post molar gestational trophoblastic neoplasia (GTN): four values or more of ß-hCG plateaued over at least 3 weeks or an increase in ß-hCG of 10% or greater for 3 or more values over at least 2 weeks. Ultrasound has become the standard protocol employed to assist in diagnosis of suspected GTN. Brightness-mode ultrasound is useful to detect the presence of abnormal uterine masses. Sonographically, an invasive hydatidiform mole typically exhibits a heterogenous, hyperechoic, solid mass with cystic vascular spaces, located within the myometrium. Color Doppler imaging aids in the assessment of angiogenesis and neovascularization characteristic in these tumors, seen as prominent blood flow signals in various directions, suggestive of arterial and venous flow. Demonstration of a vascular mass within the myometrium on ultrasonography, without evidence of fetal material, in the context of an elevated ß-hCG is highly suggestive of GTN. Its pathology is characterized by the presence of edematous chorionic villi with trophoblastic proliferation that invades into the myometrium of the uterus.  Invasive moles are rarely metastatic, but if metastasis does occur, it is usually to the lungs. Management of an invasive mole includes treatment with chemotherapy, as well as continued monitoring of ß-hCG. Patients with GTN should be followed with weekly quantitative ß-hCG levels until normal for three consecutive weeks, then monthly for 12 months. Dilatation and curettage is not recommended due to the risk of uterine perforation. With methotrexate, complete remission is achieved in most non-metastatic and low risk cases. One study reported laparoscopic removal of a corneal invasive mole and the patient was administered methotrexate chemotherapy. After laparoscopic myometrial resection, pregnancy should be avoided for at least 6 months, and a cesarean section is necessary. The value of this case report can be seen in the following three points. First, in general, more than 95% of invasive moles occur after a previous mole removal, and very rarely occur after a missed abortion. Second, in the absence of distant metastasis, treatment with methotrexate is appropriate. However, the patient had severe adverse drug reactions, pelviscopic resection of the invasive mole site for future pregnancy. Third, 1 year later, a natural pregnancy occurred and a cesarean section was performed at 36 weeks without uterine rupture. No such case has ever been reported. It was accurately diagnosed by transvaginal ultrasound and color Doppler, and successfully treated by pelviscopic resection before any major complications could arise. We suggest that this rare valuable case is likely to be of help to many clinicians.Invasive hydatidiform moles develop in approximately 10% to 20% of patients after molar evacuation and infrequently after other gestations. Due to their aggressive growth characteristics, invasive moles are considered locally invasive non-metastatic neoplasms. They are defined as moles that penetrate and may even perforate the uterine wall. The tumor is locally destructive and may invade parametrial tissue and blood vessels, eventually leading to hemoperitoneum. Conceptualization: Yun Sook Kim.Data curation: Yun Sook Kim.Investigation: Yun Sook Kim.Writing – original draft: Yun Sook Kim.Writing – review & editing: Hyun Joo Lee.Yun Sook Kim orcid: 0000-0001-8427-4006.


A 56-year-old female presented with a complaint of left flank pain for two weeks. Her past medical history was unremarkable. A left pelvic calcification was observed on abdominal X-ray . UltrasoOvarian MCT is a cystic or solid tumor  which accounts for 10-20% of all ovarian tumors , but theEarly diagnosis and treatment in terms of a conservative surgical approach is recommended. Ovarian MCT should be considered in the differential diagnosis of distal ureteric obstruction causing proximal hydroureteronephrosis in young female patients .


Inferior mesenteric arterioportal fistula (APF) is rare as only 35 case reports in the literature. We herein presented a case of simultaneously double cancer in the rectum and stomach with inferior mesenteric APF, which is the first case report by searching using PubMed. Combination of interventional embolization and surgical operation seemed to be optimal treatment for avoiding postoperative complications and the curability.A 66-year-old male with epigastric pain was admitted to a practitioner. He underwent a gastroscopy with biopsy, and cancer located in the lesser curvature of the gastric cardia was found. Enhanced CT did not reveal wall thickening of the stomach and distant metastases, but several swollen lymph nodes were observed in the right cardia. In the arterial phase, dilation of inferior mesenteric vein (IMV) and superior rectal artery (SRA) were noted, which raised suspicions of an arterioportal communication. Colonoscopy revealed a type 2 rectal tumor located 12 cm from the anal verge. The histological diagnosis of well-differentiated tubular adenocarcinoma was confirmed by biopsy. At a first step, we planned to perform a radiological embolization of inflow vessels to APFs except for SRA. Additionally, we determined the interval time of 1 month between the first low anterior rectal resection and the sequential gastrectomy for the purpose of decreasing portal pressure. The postoperative course was uneventful without hemorrhagic complications, and S-1 was taken internally 1 year as adjuvant chemotherapy for gastric cancer. The patient still lives without recurrence of this cancer with APF and portal vein thrombosis 2.5 years after the aforementioned surgeries.Inferior mesenteric APF and/or arteriovenous fistula (AVF) would be consisted of the several inflow arteries as superior rectal, internal iliac, and median sacral arteries, and outflow veins as inferior mesenteric, internal iliac, and median sacral veins. To determine the therapeutic strategy for left-sided colorectal cancers with abnormal vessel communications of the pelvis, it is significant to comprehend distribution and component vessels of APF and/or AVF. Arterioportal fistula (APF) was firstly confirmed by Weigert  in 1886.A 66-year-old male with epigastric pain was admitted to a practitioner. He underwent a gastroscopy with biopsy, and cancer located in the lesser curvature of the gastric cardia was found. The patient was transferred to our hospital for surgical treatment. He was in good health except for mild obesity (body mass index of 27) and a history of appendectomy. Abdominal examination revealed tenderness in the epigastric fossa. However, no palpable pulsatile mass was confirmed. There was no symptom of hypertension and heart failure. Laboratory findings were unremarkable except for high hemoglobin A1c levels as 6.7% (ranging 0–6%). Ultrasonic cardiography showed no remarkable right heart load and pulmonary hypertension.Upper gastrointestinal endoscopy showed an irregular ulcerated lesion with fold convergence, encroachment, and poor dispensability in the lesser curvature of the cardia, which was diagnosed as submucosal or muscular proprial infiltration by gastric cancer. Histological diagnosis revealed a moderately-to-poorly differentiated adenocarcinoma. Neither varices nor portal hypertensive gastropathy was remarkable.Enhanced CT did not reveal wall thickening of the stomach and distant metastases, but several swollen lymph nodes were observed in the right cardia. In the arterial phase, dilation of the inferior mesenteric vein (IMV) and superior rectal artery (SRA) were noted, which raised suspicions of an arterioportal communication  and the SRA Fig. ). FurtheFour-dimensional CT angiography demonstrated that the tortuous and dilated IMV supplied blood flow through several fistulas including the SRA, bilateral internal iliac artery (IIA), and median sacral artery Fig. . AbdominThese abnormal vessel communications were diagnosed as APFs which used the SRA, bilateral IIA, and median sacral artery as inflow vessels and the IMV as outflow vessel. Additionally, the synchronous double cancer was diagnosed as gastric cancer  and rectal cancer  using the Union for International Cancer Control’s classification . We wereFistula embolization was performed for the branches of the median sacral artery and bilateral IIA using a micro coil, gelatin sponge, and n-butyl-2-cyanoacrylate Fig. . Post-trAbdominal CT performed on postoperative day 14 showed a small fluid collection with gas around the anastomosis, which suggested an anastomotic leakage and thrombosis, which emerged in the right branch of the portal vein and remnant IMV. After intake of warfarin for 2 weeks, thrombotic size was markedly decreased, and the diameter of portal vein diminished in size compared to its preoperative state Fig. . The 1-wThe postoperative course was uneventful without hemorrhagic complications, and S-1 was taken internally 1 year as adjuvant chemotherapy for gastric cancer. The patient still lives without recurrence of this cancer with APF and portal vein thrombosis 2.5 years after the aforementioned surgeries.APF involving the IMV is extremely rare, and to our knowledge, only 35 cases have been reported in the literature to date –36. ArteAPFs usually occur secondarily to previous surgeries, trauma, neoplasms, and rupture of an arterial aneurysm into the adjacent vein . Other AThe presenting symptom in the majority of cases included abdominal pain and mass. It has been hypothesized that increased blood flow through APF causes portal venous hypertension, and a decreased arterial flow to the bowel wall results in non-occlusive ischemic colitis secondary to steal phenomenon. Clinical sign and symptoms were attributed to either portal hypertension including splenomegaly, ascites, and bleeding esophageal varices or ischemic colitis, which included diarrhea and lower gastrointestinal bleeding. In this patient, although no symptoms of portal hypertension and ischemic colitis were observed in the preoperative assessment, the reduced size of the portal vein detected in the postoperative CT demonstrated occult portal hypertension. Portal hypertension accompanies 42% of APF cases due to an increase of outflow and intrahepatic vascular resistance  noted thDirect angiography is the most important modality for the diagnosis and crafting strategy for APF . InferioThe treatment of APF is mainly performed by the intra-arterial embolization, surgery, and these combined procedures, and the approximately 90% of AVM cases are treated regardless of embolization or surgical resection . The embIn this case, we could not assess the existence of APFs in mesorectum with pathological examination, because of taking priority over assessment for dissected lymph nodes, regrettably. We guessed that APFs was located in the mesorectum because postoperative CT did not show APFs.We considered that median sacral artery flowed into APFs without going through middle rectal artery from the findings of flow void in the preoperative MRI. On the other hand, the possibility that bilateral IIA contributed to the constitution of APFs via middle rectal artery cannot be denied. So, we cannot affirm the validity and efficiency of bilateral IIA embolization in this case, as compared to sacral artery embolization.As described above, our present case is extremely rare because it entailed a double cancer with the inferior mesenteric APF. We were eventually able to treat it without bleeding complication by preceding IVR for APF. Two cases in the left-side colorectal cancer with comorbid abnormal vessel communications in pelvis are reported in Table We reported a rare case of simultaneous rectal and gastric carcinomas accompanied with inferior mesenteric arterioportal fistula. To determine therapeutic strategies for left-sided colorectal cancer with abnormal vessel communications of the pelvis, it is vital to understand the distribution and component vessels of inferior APF in order to make proper decisions regarding preoperative APF treatments.


Rhabdomyosarcoma (RMS), especially primary pulmonary RMS, is an extremely rare type of soft tissue sarcoma in adults. Small bowel is an uncommon site for metastases.This report described an unusual case of jejunum metastasis from primary pulmonary RMS causing intussusception in a 75-year-old man. The patient consulted for 2 weeks of continuous dyspnea. Chest computed tomography (CT) demonstrated a large mass involving the left lower lobe. Transthoracic biopsy confirmed the existence of pleomorphic RMS. Immunohistochemical studies showed positive findings about desmin and MyoD1. The results of gastroscopy, colonoscopy and abdominal CT were all negative. Positron emission tomography/CT demonstrated a fluorodeoxyglucose-reactive large lesion in the left lower lobe without metastatic lesions. The patient received synchronous chemoradiotherapy. After 9 months, the patient presented with intermittent upper abdominal pain with nausea and vomiting. CT showed small bowel dilatation secondary to intussusception. The patient subsequently received laparotomy, and the intussuscepted small bowel segment was resected. Histological examination revealed pleomorphic RMS involving the mucosa, submucosa, and muscular tissues.RMS is highly aggressive and metastatic. The metastatic disease can rapidly progress to cause subsequent complications. The possibility of small bowel metastasis should be considered, although it is extremely rare. Rhabdomyosarcoma (RMS) is a malignant skeletal muscle neoplasm. RMS is a common soft tissue sarcoma that occurs during childhood and adolescence, and it exists in the first 2 decades of life. By contrast, RMS is remarkably rare in adults. RMS accounts for < 1% of adult malignancies and < 3% of adult soft tissue sarcomas . PrimaryThe subject was a 75-year-old man with chronic obstructive pulmonary disease for more than 20 years. The patient was a nonsmoker and had no history of other remarkable illnesses. The patient consulted due to 2 weeks of continuous dyspnea without fever, cough, hemoptysis, or chest pain. Lung auscultation revealed no wheezing sounds. Laboratory examinations, including complete blood count, electrolytes, renal function, liver function, and urinalysis, were negative. Chest computed tomography (CT) confirmed the presence of a large mass lesion of 5.8 cm × 4.4 cm that involves the left lower lobe Fig. a and b. After 9 months, the patient was referred again to our hospital because of intermittent upper abdominal pain with nausea and vomiting. Physical examination showed that a large mass was palpable in the left abdomen. Bowel sounds were active. Laboratory data showed 2.1 mmol/L serum potassium. Abdominal CT showed small bowel dilatation secondary to entero-enteric intussusception Fig. a and b. RMS is a common soft tissue sarcoma in the pediatric population. RMS is initially believed to originate from skeletal muscles. However, RMS is currently known to arise from early rhabdomyoblasts that can be found in various locations throughout the body with varying degrees of differentiation . RMS thaApproximately 20% of patients have distant metastases upon presentation, which is commonly found in lungs, bones, bone marrows, lymph nodes, and rare metastatic sites –13. MetaThe incidence of small bowel metastases is low, and misdiagnosis and missed diagnosis easily occur due to nonspecific symptoms. We reported the second case of small bowel intussusception caused by PPR metastasis, suggesting that the possibility of metastasis should be considered regardless of the rarity or site location . Intussu