The falciform ligament is a double peritoneal fold that separates the left and right hepatic lobes anatomically. Fatty-falciform ligament appendage torsion (F-FLAT) is defined as torsion of the extraperitoneal fat within the falciform ligament causing fat infarction, which is an uncommon surgical presentation, scarcely documented within the current literature. The objective of presenting this case report and reviewing the literature on F-FLAT is to discuss the clinical presentation, possible associated factors and management strategies in regard to this rare pathology.A 72-year-old female patient presented to the emergency department with a seven-day history of epigastric pain, reduced appetite and nausea. On admission, the patient was stable and apyrexial with abdominal examination highlighting she was tender in her right upper quadrant and epigastric region. Due to the patient's unremitting abdominal pain despite appropriate analgesia, CT of the abdomen and pelvis (CTAP) with intravenous contrast was done and a diagnosis of F-FLAT was made. The patient was treated with antibiotics and analgesia, had a negative abdominal ultrasound (US) result and due to her symptoms settling by the second day of admission, she was discharged the same afternoon.A literature review into falciform ligament infarction was conducted by two independent reviewers across four different databases: PubMed, Medline, Embase and the Cochrane Library. Search terms included “falciform ligament” OR “falciform” AND “infarction” . Eligibility criteria and our subsequent inclusion criteria were based on studies specifically discussing falciform ligament infarction and published in English. Study types were by majority case reports, but also included one literature review and a book source as well as two pictorial radiological reviews. All 13 patients presented with abdominal pain, but only 53% presented with raised infective/inflammatory markers. The majority of patients had abdominal US as a first-line investigation with 9 of 13 patients also having a CTAP with contrast, which classically showed fat stranding in the falciform ligament. Two patients had no evidence of any radiological investigation. Initially all cases were managed conservatively with non-steroidal anti-inflammatory drugs and analgesia, but in 62% of the cases (8/13), surgical intervention was needed due to unresolving abdominal pain. All eight of the excised falciform ligaments showed evidence of infarction and necrosis histologically.In conclusion, F-FLAT is a relatively rare condition making it difficult to build higher level evidence studies. The current literature has revealed some evidence of incomplete and inconsistent data, for example, in the biochemical results and management techniques presented, yet contrast-enhanced CT seems moderately sensitive for detection in the reviewed literature. Though F-FLAT is rare and unfamiliar, it is vital we exclude common acute surgical pathologies that F-FLAT mimics and monitor for unsettling symptoms that could change the management trajectory. The falciform ligament is a double peritoneal fold that separates the left and right hepatic lobes anatomically. Fatty-falciform ligament appendage torsion (F-FLAT) is defined as torsion of the extraperitoneal fat within the falciform ligament causing fat infarction, which is an uncommon surgical presentation, scarcely documented within the current literature . It is aA 72-year-old female patient presented to the emergency department with a seven-day history of epigastric pain, reduced appetite and nausea. Pain was colicky in nature, severe in intensity and worse on food intake. There was no history of trauma or recent travel to foreign countries; there were no excess alcohol episodes or new changes in diet. She denied any other concerns or any systemic symptoms or significant co-morbidities.On admission, the patient was stable and apyrexial. On abdominal examination, she was tender in her right upper quadrant and epigastric region. Clinically, Murphy’s sign was positive, but otherwise there were no overt signs of peritonitis. The laboratory results only showed a mild raise in the C-reactive protein  level, but otherwise all other tests  were within the normal range. An erect chest x-ray ruled out a pneumoperitoneum and also excluded any chest pathology. Given that the patient kept experiencing unremitting abdominal pain despite appropriate analgesia, a CT scan of the abdomen and pelvis (CTAP) with intravenous contrast was done, and with this, a diagnosis of F-FLAT was made Figures , 2.During the admission, the patient had intravenous antibiotics for two days (a 100 mg loading dose of tigecycline followed by a single dose of 50 mg tigecycline the next day), and analgesia in the form of opioids and antiemetics.The next day, abdominal ultrasound (US) was done given her right upper quadrant (RUQ) pain and positive Murphy’s sign to rule out gallstone pathology or evidence of cholecystitis; this showed a small area of increased echotexture (fat sparing) surrounding the gallbladder fossa with no evidence of gallstones, nor any biliary tree/intrahepatic ductal dilatation Figure . The patReview methodologyA literature review was conducted by two independent reviewers across four different databases: PubMed, Medline, Embase and the Cochrane Library in July 2022. Search terms included “falciform ligament” OR “falciform” AND “infarction” . Inclusion criteria were studies with adults only, studies specifically discussing falciform ligament infarction/F-FLAT and published in English. We also excluded patients less than 18 years of age. A Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) flow diagram is included to pictorially represent the inclusion and exclusion process , inflammatory markers were raised, but due to incompletion, valid conclusions were hard to draw Table .Investigations and Diagnostic ProceduresAbdominal US was the first-line investigation for all the studies reviewed except two Table . In thesManagement: Conservative Versus Surgical Figure Surgical Follow-Up and Histology ResultsAll the surgically managed patients had intraoperative samples from the excised ligament sent to pathology. Histology results showed fat tissues, high vascularity and inflammation and necrotic, thrombotic and inflammatory infiltrated cells of the resected tissue. Furthermore, only four studies had a documented follow-up, with two studies reviewing in two weeks post-diagnosis, one study following up eight weeks after and one with no timeframe reported. All four patients in the studies had completely resolved symptoms.Interpretation of findingsInfarction of the falciform ligament, pathologically defined as F-FLAT, is a rare diagnosis, with only 13 patients having F-FLAT reported in the literature till now. Other types of IFFI such as omental infarction and epiploic appendagitis are more common , yet areThe falciform ligament is derived embryologically from the ventral mesentery and thus attaches from the ventral abdominal wall and inferior diaphragmatic surface, stretching past the anterior liver margin and across to the posterior visceral surface. This consequently gives a clear demarcation of the liver’s left medial and right lateral lobe. Arterial drainage comes from the middle hepatic arteries and left inferior phrenic artery whereas the left inferior phrenic vein is the sole venous drainage. The ligament’s free edge consists of the ligamentum teres, obliterated umbilical vein  and extraperitoenal fat/appendages, the latter of which can vary in individuals across the whole ligament’s tract, and where F-FLAT is pathologically defined . It occuThe population group and the clinical presentation were the first area we broke down: we found that over 80% of patients were adult females, but it is difficult to associate the two sexes with the pathology, due to the condition being so rare. The review also illustrated that past medical and surgical history of the patients were scarcely documented, with only three cases giving evidence of them, which makes any specific risk factors almost impossible to correlate and justify. Presentation wise, every case study documented abdominal pain as the primary symptom followed by nausea as the second most common; pain in over half of the cases reviewed was localised to the epigastric and right upper quadrant area. It has been described in the literature that this acute presentation can mimic other surgical pathologies such as appendicitis, cholecystitis and less commonly, hepatitis and bowel perforation -9.Biochemically, there was some evidence of mildly raised inflammatory markers, yet 50% of patients had a normal white cell count and CRP levels. Moreover, both these were documented in only six studies as there were numerous data gaps within the blood results reviewed, further illustrating insufficient data. Imaging is therefore crucial in diagnosing F-FLAT.Ultrasound typically presents with a hyperechoic mass with adjacent inflammatory changes, heterogeneous in size and noncompressible, deep to the rectus abdominis, has absent colour flow on Doppler and corresponds to the patient's pain on palpation ,11. LessAbdominal CT with contrast is much more sensitive with coronal and sagittal views depicting increased density within the fat joining the falciform ligament with associated inflammatory changes as well as sometimes a peripheral rim of hyperattenuation, with the “hyperattenuating rim sign” indicating vascular compromise. Furthermore, one can subsequently see “a central dot sign” that is a hyperdense focus indicating a thrombosed central vein . Out of A conservative management approach to F-FLAT is preferred, yet due to a worsening clinical picture and unresolving abdominal pain, surgical resection might be indicated. Analgesia (NSAIDs/opioids) was the most common conservative method used, and specific antibiotic use was not mentioned in any of the F-FLAT cases we identified. Furthermore, other than the one laparotomy case, surgical techniques and details were not expanded on in the rest of the cases we reviewed, and data were minimal. Due to the condition being so rarely reported in the literature and the limited data available, one cannot compare conservative versus surgical management at first presentation, but can infer a conservative approach is preferable in the initial F-FLAT presentation.F-FLAT is a rare and unfamiliar presentation that warrants careful attention to differentiate it from other common acute pathologies that it can imitate. The literature review of this rare condition suggests that F-FLAT occurs more commonly in adult females and always presents with abdominal pain, usually epigastric or in the RUQ region. Biochemical markers are nonspecific, and thus imaging with CTAP with contrast is often required to diagnose this rare condition and also exclude common acute surgical pathologies that F-FLAT tends to mimic. Initial management with analgesics alone is not unreasonable, though surgical resection with a laparoscopic approach where available might need to be done if symptoms do not settle, which could be a marker for necrosis.


A urachal mass is a relatively rare presentation to the urologists’ practice, often requiring radical surgical excision for a definitive diagnosis. Xanthogranulomatous inflammation of the urachus is an extremely rare entity with few cases reported worldwide, and to the best of our knowledge, no cases reported in the western world.In this case, a 55-year-old male patient presented with bothersome lower urinary tract symptoms and computed tomography findings demonstrating a urachal mass that was worrisome for urachal carcinoma. Following surgical intervention, histopathology revealed urachal xanthogranuloma. Post-operatively, the patient recovered well, and eventually, he had symptomatic and radiologic improvement.This case brings awareness to a rare presentation of a urachal mass—urachal xanthogranuloma. While operative intervention was both diagnostic and therapeutic, we highlight the challenge in differentiating between benign and malignant processes for urachal masses. Herein, we show the importance of including urachal xanthogranuloma in the differential diagnosis of a urachal mass to prevent further morbidity associated with the treatment of this disease. Xanthogranulomatous disease is a form of chronic inflammation. Xanthogranulomatous inflammation of the urachus is an extremely rare entity with few cases reported worldwide, and to the best of our knowledge, no cases reported in the western world. Given the difficulty of distinguishing between carcinoma and xanthogranuloma on imaging or frozen section and the high rate of infection in granulomatous tissue, diagnosis is made by surgical excision and histopathologic examination , 2.Herein, we present a 55-year-old male with a suspected urachal carcinoma. Following surgical excision, this mass was diagnosed as urachal xanthogranuloma.A 55-year-old Argentinian male was referred to a Canadian urologist’s practice in February 2021 with a three-month history of left lower abdominal pain, urinary frequency, nocturia, and urgency. The patient denied bothersome obstructive lower urinary tract symptoms, gross hematuria, dysuria, urinary incontinence, and B-symptoms including weight loss and change in appetite. His past medical history was significant for obesity and a laparoscopic cholecystectomy in 2017. He had no known drug allergies or medications. Physical examination demonstrated a patient with truncal obesity, weight of 125 kg, height of 178 cm, no abdominal masses, unremarkable umbilicus, unremarkable male genitalia, and a benign prostate. Screening hematologic, extended electrolytes, creatinine, blood urea nitrogen, glomerular filtration rate, and prostate-specific antigen levels were unremarkable. Urinalysis, urine culture, and urine cytology were negative. Abdominal ultrasound demonstrated an unremarkable post-void residual and prostate size but an abnormal 4.2 × 6.1 × 5.3 cm mass near the superior aspect of the bladder, prompting further investigation. Cystoscopy was unremarkable and did not reveal a bulge at the dome. Computed tomography (CT) abdomen/pelvis revealed a 10.3 cm solid and cystic mass abutting the superior bladder and extending up to the level of the umbilicus. There was associated mild soft tissue thickening, stranding, and nodularity on the anterior aspect of retro-umbilical omentum (Fig. In March 2021, the patient underwent an open surgical excision of a urachal mass, partial cystectomy, and bilateral extended pelvic lymph node dissection. Gross pathology revealed a 14.5 × 13 × 9.5 cm mass abutting the superior aspect of the bladder (Fig. Post-operatively, the patient had an uncomplicated hospital course. His two-week post-operative CT cystogram did not reveal a bladder leak; thus, his foley was discontinued and the patient passed his trial of void. At his two-month follow-up visit, he had returned to his baseline health with resolution of his urinary symptoms. Follow-up imaging revealed the absence of the urachal mass and no further worrisome findings.The urachus is an embryological remnant of the allantois that normally obliterates during the 5th to 7th month of gestation to ultimately form the median umbilical ligament . It is r.Xanthogranulomatous inflammation is pathologically characterized by the accumulation of lipid-laden foamy macrophages. While various sites may be affected, xanthogranulomatous pyelonephritis is the most common urologic presentation. Urachal xanthogranulomatous inflammation is exceedingly rare worldwide with no known reported cases in North America and few reported cases from Japan, Korea, and Spain .CT is used as an initial diagnostic modality to help diagnose urachal masses. CT findings of urachal xanthogranuloma often reveal a mass that is characterized as cystic, solid, or a solid-cystic lesion in the dome of the bladder , 6. DiffWhile no guidelines exist to support clinicians in managing urachal xanthogranulomas, the current consensus in managing suspected urachal carcinoma is en bloc surgical removal of the umbilicus, urachal ligament, and partial cystectomy with pelvic lymphadenectomy . This inIn conclusion, xanthogranulomatous inflammation of the urachus is a rare but important entity to include in the differential diagnosis of urachal masses. Our case study highlights the challenge in differentiating between urachal carcinoma and urachal xanthogranulomas. It is evident, not only from our case but also from the few other cases reported globally that this mass is easily misdiagnosed on imaging and requires definitive diagnosis with surgical excision and histopathology. Ultimately, surgery was both diagnostic and therapeutic; however, there is a need for earlier, more accurate diagnosis to minimize additional interventions and morbidity associated with aggressive surgical treatment. This case is especially important to urologists globally, as it will hopefully inform them about the diagnosis and management of urachal xanthogranulomas and spur advancements in differentiating it from malignant conditions.


Skin tumors are prevalent in the head and neck especially auricle but pilomatricoma is extremely rare in ear lobule.month history of a lesion in the right ear lobule that tended to grow in size. It was a 2*2*2cm round, slightly painful lesion with soft tissue and light red in color that yielded bloody or serous fluid on aspiration. The lesion was enucleated. The diagnosis was pilomatricoma. A 7-year-old girl without a history of previous illness presented with a 1.5-Although very rare, pilomatricoma should be considered as a differential diagnosis of ear lobule neoplasms. This tumor accounts for 0.12% of all skin tumors  on the lesion. The patient first visited a general practitioner and dermatologist and was treated with topical tetracycline ointment and cefixime syrup for a week, but did not respond to treatment and the lesion continued to increase in size. The patient then visited an otolaryngologist and underwent aspiration three times. The lesion yielded bloody or serous fluid on aspiration; however, it was still enlarging in size. With the possible diagnosis of colloid nodules, an intralesional injection was performed, after which the lesion growth was intensified. The patient came to our clinic with a 2*2*2cm sized round, slightly painful lesion, light red in color in the right ear lobule. right earlobe. The lesion was a soft tissue, nodule-like mass with slight tenderness on examination that yielded bloody or serous fluid on aspiration. The lesion was drained and enucleated. Pieces of tissue sections containing islets of basaloid cells and islets of shadow cells were observed in the biopsy specimen, indicating a pilomatricoma lesion. shows the patient's sample pathology report. After 9 months there were no recurrence  and matricial carcinoma ( for pilomatricoma is complete surgical excision ( should be suspected in cases where recurrence is reported. Histopathologically, arcinoma . The theexcision . But enuexcision . Malignapilomatricoma is rare in auricle and lobule, it should be taken into account in differential diagnosis of lobule masses.Although


Limited studies report on the status of surgical closure patches for atrial septal defect (ASD) in the remote period. In our case, transthoracic echocardiography revealed a fistula of ASD patch before pulmonary vein isolation for atrial fibrillation. Preoperative Imaging examinations aid in evaluating the effect of the needle puncture around the artificial material of the atrial septum and catheter manipulation for patients with a history of ASD closure. A 72‐year‐old man was referred to our department due to palpitations secondary to paroxysmal atrial fibrillation. He had undergone surgical closure of an atrial septal defect (ASD) approximately 50 years prior. Electrocardiogram revealed paroxysmal atrial fibrillation (AF). Transthoracic echocardiography (TTE) revealed dilation of the left atrium and right ventricle Figure . The rig2 score; 2 points, CHA2DS2‐VASc score; 3 points). Prior to radiofrequency catheter ablation, preoperative cardiac imaging evaluation of the ASD closure patch was planned.According to the diagnosis of AF, edoxaban , bisoprolol (0.625 mg/day), and amiodarone (50 mg/day) were prescribed  revealed an atrial shunt Figure . TransesBased on the location of the slit identified by TEE prior to the ablation procedure, we aimed a sheath to the slit using the intracardiac echocardiography (ICE) AcuNav catheter (Biosense Webster Inc) and successfully passed a guidewire through to the left atrium without a needle puncture. To avoid increasing the leakage due to manipulation of a large‐diameter sheath, an 8.5‐Fr steerable sheath was only inserted into the left atrium through the slit followed by extensive encircling pulmonary vein isolation (PVI). PVI was performed with a fast anatomical mapping image Figure . Post‐abOver 6 months of follow‐up, this patient had no recurrence of AF nor any evidence of increased leakage.Surgical closure of atrial septal defects (ASDs) has been a standard modality of treatment for decades. Since the first reports of surgical ASD closure in 1948, more than 70 years of experience have resulted in a safe and effective surgery with minimal mortality and complications. However, prolonged follow‐up is rare for patients with a history of surgical treatment for ASD. Thus, to the best of our knowledge, limited studies report the long‐term status of the closure patch.Those studies that have reported the long‐term outcomes after surgical repair of ASD generally neglect to evaluate the status and material of the closure patch.In patients with surgically repaired ASDs, the transseptal puncture method can be performed via the neighboring native atrial septum tissue or the patch itself.Here, we reported a case of ASD, detected by preoperative TTE and cardiac CT, where a transseptal approach via minor peri‐closure patch leak was used. Imaging examinations provided important indications informing puncture sites. Evaluation of the status of the closure patch before using a transseptal puncture approach in patients with a history of treatment for ASD has been emphasized, although the follow‐up data of most patients with a history of surgical closure of ASD long time ago are often unavailable because of censored follow‐up. Thus, understanding the utility and benefits of preprocedural multi‐modality imaging to assess the ASD closure patch may improve the utility of the transseptal approach.This research did not receive any specific grant from funding agencies in the public, commercial, or not‐for‐profit sectors.The authors have nothing to disclose.Approval was obtained from the local ethics committee.Written informed consent was obtained from the patient for the publication of this case report.Not applicable.Not applicable.N/A.Figure S3Click here for additional data file.Movie S1Click here for additional data file.Movie S2Click here for additional data file.


Nevus sebaceous (NS) presents as alopecia and yellowish discoloration during infantile stage. In adult stage, lesions become verrucous. Importantly, various appendageal tumors such as trichoblastoma, syringocystadenoma papilliferum and basal cell carcinoma develop during this stage. Hence it is very important to follow the course of NS for early detection of neoplasms. We are presenting a case of a 10-year-old patient with a dome-shaped, dark-pigmented nodule on the left side of neck nape, which later diagnosed as NS and removed with a carbon dioxide laser. Jadassohn first identified nevus sebaceous (NS) as a concentric hamartomatous lesion made up of sebaceous glands in 1895 . During Lesions become more verrucous and lobulated in the adult or third stage. It is important to note that during this stage, a variety of appendageal tumors, both benign and malignant, like trichoblastoma, syringocystadenoma papilliferum and basal cell carcinoma, form , 6. As aA 10-year-old healthy male presented with a dome-shaped, dark-pigmented nodule on the left side of neck nape . The pigA diagnosis of NS of the neck nape was made following the history and physical examination of the area.2) laser therapy because of the extensiveness of skin lesions and the effectiveness and accessibility of the method. One-year follow-up shows no recurrence  specially when the lesion is big to avoid big scars or skin tension-deformity-hypertrophic scars. However, because of the possibility of local relapses and malignant transformation inside residual lesions, choosing this therapeutic strategy necessitates further follow-up.2 laser therapy. However, because of the possibility of local relapses and malignant transformation inside residual lesions, choosing this therapeutic strategy necessitates further follow-up.A number of the only procedure offering a complete excision and oncologic safety for NS is surgical removal. Other recommended treatment methods for NS include photodynamic therapy, dermabrasion, laser therapy and cryotherapy. An alternative to early surgical removal of skin lesions is still clinical surveillance. Because of the patient’s significant skin lesions, as well as the treatment’s efficiency and accessibility, we decided to use CO


Vulvar leiomyomas are extremely rare smooth muscle tumors that are easily mistaken for other lesions, as the differential diagnosis must consider a wide spectrum of benign and malignant lesions. We present the case of a 52-year-old woman with a three-year history of progressive abdominal distension and pain and an enlarging vulvar mass distorting the labia majora and causing gait disturbance. Imaging confirmed an enormous pelvic mass originating in the uterus, compatible with a leiomyoma/sarcoma, and large perineal and vulvar masses with similar characteristics. Histopathology after surgical removal revealed benign abdominal, vulvar, and perineal leiomyomas. This case highlights the rarity and diagnostic challenges of extra-uterine leiomyomas, particularly those in the vulvar region. Leiomyomas are well-circumscribed, benign, smooth muscle neoplasms that most commonly present as uterine tumors in females of reproductive age. While uterine leiomyomas are fairly common, extra-uterine ones are not, and vulvar leiomyomas, in particular, are extremely rare. The unusual location and painless clinical presentation of vulvar leiomyomas present diagnostic challenges along with the fact that vulvar masses include a wide spectrum of benign and malignant tumors . UltrasoWe report the case of a 52-year-old woman presenting with abdominal pain and anemia, exuberant vulvar and perineal masses causing gait disturbance, and a concomitant abdominal mass. Surgical excision was performed, and histologic analysis confirmed uterine, vulvar, and perineal leiomyomas.A 52-year-old multiparous female presented to the gynecological emergency department with a large vulvar mass, which had been growing for three years and was associated with gait disturbance, diffuse abdominal pain, and abdominal distension. No hormonal contraceptives were used and menstrual cycles were regular. Other symptoms, such as vaginal bleeding, fever, and recent weight loss, were not present. The patient had a medical history of high blood pressure, dyslipidemia, and previous cholecystectomy.Gynecological examination revealed a voluminous vulvar mass distorting the labia majora. The mass was firm and somewhat mobile, without tenderness Figure . SpeculuAbdominal and pelvic ultrasound showed a heterogeneous mass occupying the whole pelvic and inferior abdominal cavity and a vascularized vulvar mass with similar characteristics. CT demonstrated a 24-cm pelvic mass originating in the uterus with regular contours and heterogeneous nodular enhancement, compatible with a leiomyoma/leiomyosarcoma. Numerous vulvar and perineal masses were also noted Figures .MRI showed well-demarcated uterine, vulvar, and perineal masses, with hypointensity on T2-weighted images, absent restricted diffusion, and progressive heterogeneous enhancement on T1 post-gadolinium sequences, suggesting leiomyomas, although sarcomas could not be excluded Figures , 5. The The patient underwent a total abdominal hysterectomy with a bilateral adnexectomy. Intraoperatively, the uterus was markedly enlarged (30 cm) and appeared to be totally occupied by fibroids. The right vulvar masses measuring up to 10 cm and the left perineal masses measuring up to 7 cm were also removed Figures , 7.Pathology revealed a uterus replaced by leiomyomas and vulvar and perineal leiomyomas with no atypia or considerable mitosis. At two years follow-up, the patient is well and has had no recurrence of the disease.Leiomyomas are the most common uterine neoplasms, known to affect 30% of women over the age of 35. They are benign soft tissue mesenchymal tumors derived from smooth muscle cells ,2.Leiomyomas are most often found in the uterus, with extra-uterine locations being rare. Vulvar leiomyomas account for only 0.03% of all gynecologic neoplasms and 0.07% of all vulvar tumors, with only about 300 cases described in the literature -4.Vulvar leiomyomas originate from the smooth muscle within the round ligament, erectile tissue, and blood vessel walls ,4. TheirMost patients present with a long history of a painless mass that can sometimes compress the urinary bladder and rectum . As the Ultrasound is an accessible method to confirm the location and size of leiomyomas. MRI is valuable for planning surgery and for characterizing leiomyomas’ vascularization, soft tissue invasion, and relationships to nearby organs.MRI usually shows low signal intensity like that of smooth muscle on T2-weighted images and low signal intensity in diffusion-weighted images. Leiomyosarcoma is the main concerning differential diagnosis and usually manifests as an ill-defined, heterogeneous myometrial mass that frequently presents with focal areas of high signal intensity on T1-weighted images corresponding to hemorrhage or necrosis, intermediate to high signal on T2-weighted images and low apparent diffusion coefficient (ADC) values on diffusion-weighted images. The frequent overlapping features make differential diagnosis difficult and MRI cannot safely exclude malignant transformation as 0.01%-0.5% of resected tumors diagnosed preoperatively as leiomyomas are confirmed as leiomyosarcomas .Histopathological examination is the only accurate way to verify benignity and to rule out the presence of other kinds of mesenchymal vulval tumors, which can exhibit aggressive behavior ,7. HistoSurgical excision is the preferred approach to definitive diagnosis and the mainstay of treatment, with long-term follow-up advisable to detect recurrence . DiffereExtra-uterine leiomyomas are rare, and an unusual vulvar lobe location poses challenges for diagnosis. Physical examination usually detects a firm mass, with ultrasound and MRI essential for planning surgery and facilitating differential diagnosis. Although benign, these leiomyomas can mimic aggressive mesenchymal vulvar tumors during observation and imaging. Therefore, surgical excision and histologic characterization are mandatory for confirming the diagnosis.Early diagnosis along with appropriate management and follow-up depends on thorough examination and a familiarity with their extrauterine sites and typical imaging features. Raising awareness of these uncommon lesions can also contribute to effective diagnosis and management.


Small bowel arteriovenous malformation (AVM) is a rare vascular lesion, which should be considered in patients presenting with gastrointestinal bleeding, as it is a high-flow arterial lesion that can cause life-threatening bleeding. Although a primary endoscopic examination is performed in cases of suspected small bowel bleeding, the diagnosis of the causal lesion is sometimes difficult. We are presenting a case of small bowel AVM that could not be diagnosed endoscopically but was successfully detected using multiphase CT images with an appropriate protocol. The AVM diagnosis was confirmed using digital subtraction angiography. An endovascular coil is placed in the draining vein as a surgical resection marker. The AVM was resected successfully without any complications. Small bowel bleeding constitutes the majority of obscure gastrointestinal bleeding cases , for whiA 62-year-old man with cirrhosis presented with anemia and melena. He had a history of three endoscopic injection sclerotherapy and ligation for worsening or ruptured esophageal varices within one year. His hemoglobin decreased from 13.3 g/dL to 7.6 g/dL in one year. Upper and lower endoscopies revealed no recurrence of the varices or any other bleeding lesions. Video capsule endoscopy revealed an erythematous, protruding subepithelial lesion in the mid small bowel . However, a small bowel lesion was not detected during the initial CT examination. Due to the patient's stable condition, conservative treatment with iron supplementation was continued, but his anemia did not improve, with a hemoglobin of 7.4 g/dL after four months. Thus, the multiphase CT angiography was repeated for a more detailed evaluation using the bolus-tracking method, with the region of interest set within the abdominal aorta at the level of the celiac artery, using a trigger attenuation threshold of 150 HU. The arterial-phase images were obtained after an additional trigger delay of 10 s. The portal venous- and delayed-phase images were obtained 20 and 170 s after the arterial phase, respectively. The arterial phase images revealed an abnormally enhanced lesion measuring 1 cm in size in the small bowel. The lesion was connected directly to a dilated ileal vein, suggesting the presence of a vascular lesion (Figures ,The patient was hemodynamically stable, but persistent bleeding was suspected, and invasive treatment was considered necessary. Digital subtraction angiography, including percutaneous transhepatic portography and superior mesenteric arteriography, was therefore performed to confirm the diagnosis and to embolize the lesions. The portography and balloon-occluded retrograde angiography via the draining vein did not reveal any small bowel lesions. Subsequently, the superior mesenteric arteriography demonstrated multiple tiny feeding ileal arteries supplying the lesion and early venous drainage to the ileal vein, confirming the diagnosis of an AVM Figure . SurgicaOne week later, an open partial ileal resection and manual anastomosis were successfully performed. Intraoperative fluoroscopy demonstrated the coil clearly, aiding in the localization of the lesion and the determination of the resection line. A histopathological examination confirmed the diagnosis of an ileal AVM Figure . The aneSmall bowel vascular lesions are the most common causes of obscure gastrointestinal bleeding . They arAVMs exhibit characteristic angiographic features, including dilated arteries, a nidus, and an early venous return . TherefoWe encountered a case of a small bowel AVM that caused bleeding and could not be detected in double-balloon endoscopy. Repeated multiphase CT angiography demonstrated the lesion successfully, and the subsequent digital subtraction angiography confirmed the diagnosis of AVM. Thus, multiphase CT angiography using an appropriate protocol can serve as a salvage diagnostic examination method. Endovascular coil placement following diagnostic angiography is useful for localizing lesions during surgical resection.


Intravascular myopericytomas are a rare type of myopericytomas. In most previously reported cases, these were benign, occurred on the legs or neck, and had low recurrence rates. We have described a unique case of an intravascular myopericytoma that caused spontaneous deep vein thrombosis.A 37-year-old man presented with sudden-onset pain and swelling in the upper arm; physical examination revealed a 10 cm, palpable, firm, and mobile lesion in the upper arm. A biopsy revealed intravascular myopericytoma; immunohistological examination revealed a lesion in the lumen of the basilic vein. The tumor comprised abundant myxoid stroma with spindle cells proliferating in a concentric perivascular manner around the blood vessel. The tumor cells stained positive for CD34 and smooth muscle actin.The patient underwent total excision of the mass under local anesthesia; no recurrence was observed thereafter. A literature review was performed using PubMed and Google Scholar; the key terms were “intravascular myopericytoma” and “IVMP.” Nineteen cases of intravascular myopericytomas across 14 articles published between January 2002 and January 2022 were identified. These involved 11 men and 7 women (sex was unknown in 1 case); the ages were 22 to 80 years (mean: 59.8 ± 14 years). In most cases, the tumor was slow-growing, and the etiology was previous surgical history or trauma. No pain was reported by patients with tumors on the face or feet, and no recurrence was observed after surgery in any of the reported cases. Immunohistochemical staining for smooth muscle actin, h-caldesmon, calponin, and CD34 was performed for differential diagnosis. Contrary to the slow-growing nature reported in the literature, the nature related to growing in the present case was unclear that lesion was discovered because of sudden pain caused by thrombosis. However, the diagnostic method and recurrence rate in our case were similar to those in the previously reported cases.Our case shows that although intravascular myopericytomas are rare, they can cause spontaneous thrombosis. They have low recurrence rates after complete resection. Spontaneous deep vein thrombosis that occurs in rare locations must be treated after determining the causes. In 1992, Dictor et al defined myopericytes as transitional cells between smooth muscle cells and pericytes in the blood vessels. Myopericytoma was officially classified as a subtype of perivascular tumors by the World Health Organization in 2002. Although several reports and literature reviews on myopericytomas have been published, <20 cases of intravascular myopericytomas have been described since the first report by McMenamin and Calonje in 2002. In this report, we have presented a case of an intravascular myopericytoma in the upper arm that was discovered because of the occurrence of spontaneous thrombosis due to luminal narrowing caused by intravascular myopericytoma. We have also presented our findings from a literature review of previously reported cases.Myopericytomas, first described by Granter et alA 37-year-old man visited our outpatient clinic with complaints of pain and swelling in the upper arm, which developed suddenly 1 day before presentation.The patient used to take antithrombotic drugs for excessive obesity and cardiac hypertrophy in the past but was not taking them currently. To lose weight, he had undergone a gastric sleeve resection 4 months prior to presentation, which resulted in a weight loss of approximately 30 kg; he had maintained this. The patient denied any history of trauma.Grossly, a non-erythematous lump was observed on the inner side of the upper arm. It was tender upon palpation during physical examination and was found to be a firm but slightly movable mass-like lesion (approximately 10 cm in size). These observations suggested an intravenous thrombus rather than a lipoma or cyst. The timeline of the case is shown in Figure Ultrasonography was performed to differentiate between potential causes of the lump, such as lipomas and cysts. Magnetic resonance imaging, performed for a more accurate diagnosis, revealed that the basilic vein was enlarged and filled with a thrombus Fig. . Non-vitThe tumor  was visible on hematoxylin–eosin-stained sections (×400) during a histopathological examination. Concentric perivascular proliferation of bland, oval-to-spindle cells was observed along with multi-layered enveloping of the blood vessel . Immunohistochemical staining was negative for S-100 (positive in neurogenic tumors), desmin (positive in myogenic tumors), and smooth muscle actin and CD34 .Thus, the patient was diagnosed with an intravascular myopericytoma based on the histological examination results.The patient recovered well and experienced no complications, such as pain, in the first 7 post-operative days. Subsequently, all medications administered during treatment were discontinued. No recurrence was observed on physical examination or ultrasonography at 1 year after the surgery.We conducted a literature search in PubMed and Google Scholar using the key terms “intravascular myopericytoma” and “IVMP.” This yielded 14 case reports on intravascular myopericytomas published between January 2002 and January 2022; 1 of these was also mentioned in a literature review on myopericytomas. Although histopathological, etiological, and recurrence data were not available in the literature, we were able to collect information on patient sex, age, and location. proposed that pericytes (perivascular modified smooth muscle cells) are the origin of hemangiopericytomas. Their theory was verified by studies analyzing the structural patterns of these tumors,–8 but not fully supported by immunohistochemical findings. Staining has been performed nonspecifically for vimentin and CD34, but not for actin or other myoid markers.–10 On the basis of their clinical features, hemangiopericytomas are classified as adult hemangiopericytomas, sinonasal hemangiopericytomas, infantile hemangiopericytomas, and myopericytomas. Myopericytomas are closely related to myofibromas, glomus tumors, and angioleiomyomas; however, their distinctive feature is the presence of perivascular, concentric, multi-layered, oval-to-spindle cells. Most of these tumors are benign,13; however, multiple malignant tumors may develop and have been found to be related to the acquired immunodeficiency syndrome.,15In 1942, Stout and MurrayThe main limitation of the present case report is that it does not present any unique laboratory values; however, it has described a case involving spontaneous deep vein thrombosis, a previously unreported clinical feature of intravascular myopericytomas. In this report, the post-surgery follow-up period was approximately 1 year. Therefore, recurrence could not be confirmed through an extended follow-up period. The misdiagnosis and treatment of this condition as simple deep vein thrombosis rather than deep vein thrombosis caused by IVMP , might lead to potential recurrence. Therefore, this case emphasizes the necessity of confirming the diagnosis through complete surgical resection and a pathological biopsy.Based on our literature review, this is the first case of a myopericytoma presenting with thrombosis, which was discovered incidentally and without any history of trauma or surgery. In the basilic vein where the intravascular myopericytoma occurred, a venous aneurysm was formed due to the obstructed blood flow; this likely induced the thrombosis. conducted a histopathological retrospective review of myopericytomas and found that the mean age of the patients was 59.2 ± 14 years, with slightly more cases reported in men than in women (11 vs 6). Compared to myopericytomas, which occur at an average age of 52 years, intravascular myopericytomas appear to occur at an older age. However, intravascular myopericytomas most frequently occur in the extremities , followed by the neck and head; thus far, the present case is the only 1 involving the upper arm.As shown in Table ,16–22 Tumors that occur on the face and feet have rarely been reported to be painful.–27 McMenamin and Calonje assumed that pain is caused by a tumor thrombus, as in our case. Furthermore, in all the previously reported cases, the etiology has been attributed to previous surgical manipulation or trauma.,24,25,27Myopericytomas are not accompanied by pain; however, most intravascular myopericytomas are characterized by slow growth (8 months–30 years) and pain, which are reported in about half of the cases. Vimentin is used to identify tumors of mesenchymal origin, but is not widely used due to its non-specificity. CD34 distinguishes hemangiopericytomas (positive staining) from endometrial stromal sarcomas (negative staining). In our case, staining was performed for smooth muscle actin and CD34 (common markers used for diagnosis); staining for desmin and S-100 was performed to rule out tumors of other origins.In most cases, immunohistochemical staining was performed for smooth muscle actin, followed by h-caldesmon. CD34, calponin, vimentin, and Masson trichrome staining were rarely performed. Because h-caldesmon stains smooth muscle cells and calponin stains actin filaments, they are used to identify tumors originating from smooth muscle cells.In all cases of intravascular myopericytomas in the literature as well as in our case, the tumors were diagnosed as benign, and no recurrence occurred after surgical resection. These observations were contrary to those in previously reported cases of myopericytomas in general, wherein recurrence occurred in 5% to 10% of the cases.Our report identifies spontaneous deep vein thrombosis as a rare complication of intravascular myopericytomas, which may help in their detection. These tumors have low recurrence rates, and complete resection is an effective treatment.The patient was satisfied that they had no recurrence and no discomfort, except for mild postoperative pain.We would like to thank Editage for English language editing.Conceptualization: Kwon Cheol Yoo.Data curation: Kwon Cheol Yoo.Formal analysis: Kwon Cheol Yoo.Investigation: Lee Chan Jang.Methodology: Lee Chan Jang, Kwon Cheol Yoo.Project administration: Kwon Cheol Yoo.Supervision: Lee Chan Jang.Writing – original draft: Kwon Cheol Yoo.Writing – review & editing: Kwon Cheol Yoo.


A protostylid is a relatively rare dental developmental aberration characterized as an extra cusp located on the mesial half of the buccal surface of the molars. A protostylid is rarely to be reported due to its low rate of occurrence. This case report describes a patient referred for endodontic treatment due to the presence of a protostylid on the buccal surface of the maxillary first molar that induced apical periodontitis.A 53-year-old female reported a 3-month history of pain of chewing with her upper left posterior teeth over 3 months. In the clinical examination, an abraded anomalous cusp-like structure was found on the buccal surface of tooth 26, Cone beam computed tomography (CBCT) revealed a supernumerary cusp with an intact root canal inside, which was fused with the mesiobuccal (MB) root canal in the middle of the root. In addition, extensive periapical radiolucency was observed around tooth 26. The tooth was diagnosed as apical periodontitis, and endodontic treatment was performed. The initial lesion in tooth 26 gradually healed over 1 year of observation.To our knowledge, this case is the first to describe the endodontic management of a maxillary first molar with a protostylid and advances our understanding of supernumerary cusps. This case provides a reference for the treatment of protostylid. The protostylid is a supernumerary cusp located on the mesial half of the buccal surface of the molars , 2. The The aetiology of protostylid formation remains unknown, but there is a multifactorial aetiology that includes genetic and environmental factors , 7. ReceProtostylid is rare to be reported, and the fusion of the mesial buccal cusp with a protostylid associated with root canal treatment has not yet been reported. Thus, endodontic management of a protostylid associated with the maxillary first molar is reported in this case.A 53-year-old female patient reported a chief complaint of 3 months of pain upon chewing on her upper left posterior teeth. There was no history of trauma or any hereditary conditions. The medical history of the patient was noncontributory. An abraded anomalous cusp-like structure on the buccal surface of tooth 26 that extended from the cervical edge of the tooth towards the mesiobuccal cusp was discovered during an oral cavity examination Fig. a, b. In On radiographic examination, three roots that were generally normal in shape and size were identified, and extensive periapical radiolucency was observed around tooth 26 Fig. c. In addAn endodontic access cavity was prepared on the occlusal surface using a fissure bur and a No. 2 round bur. The tooth was isolated immediately upon the discovert of the root canal, following which root canal therapy was conducted. Next, the pulp chamber was examined under 8 × magnification by an operational microscope . Interestingly, we found that it had 5 root canals: the mesiobuccal (MB) canal, the second mesiobuccal (MB2) canal, the distalbuccal (DB) canal, the palatal (P) canal and the root canal in the protostylid, which is fused with the MB root canal in the middle of the root . The root canal was dried with paper points and filled with Vitapex paste . Afterwards, the access was sealed with Cavit .The patient was called back after 3 months for obturation. Although the clinical examination, it was found that the tooth was functioning and showed no symptoms of oedema. During the appointment, the Vitapex paste was removed using 1% NaOCl, and the root canals were sealed with the iRoot SP root canal sealant  and warm gutta-percha using a vertical condensation technique Fig. a. CBCT b and perThe patient was scheduled for follow-up visits and seen again at 6 months and 1 year after treatment. At the 6-month follow-up, a periapical radiograph was taken, and the image revealed a reduced apical area Fig. d. At theHuman teeth may exhibit various changes in morphological features. Developmental factors determine how teeth develop, particularly the form of new cusps. Thus, initiation and placement of presumptive cusp tips, epithelial folding and mesenchymal growth, termination of crown formation, and initiation of root formation are all factors that determine the shape of the tooth crown .A supernumerary cusp is an abnormality of the tooth shape. A supernumerary cusp usually refers to an accessory cusp on the buccal or lingual surface of a normal crown such as a talon cusp, central cusp or Carabelli cusp , 12. In The aetiology of protostylid remains unknown, but various theories have been proposed to explain their existence. Some researchers have hypothesized that protostylid originate from the growing centre of an enamel knot that will eventually become the location of the cuspid vertices during the morphogenetic phase, which is before the onset of amelogenesis and dentinogenesis , 14.It should be noted that the diagnosis of a protostylid should be determined carefully because it is difficult to be differentiate between a fused tooth and a concrescent tooth. Generally, fusion results from the union of two or more developing contiguous dental germs at the enamel, dentin, and cementum, which is usually observed in primary and permanent dentition and particularly appears in the anterior position , 16. A cThe management of supernumerary cusps is diverse and mainly determined on an individual basis, which includes no treatment, sequential grinding, pit and fissure sealants, pulp therapy, restorative treatment, full crown coverage, and extraction of the affected tooth . ProtostIn conclusion, the occurrence of protostylid is a relatively rare phenomenon. To our knowledge, this case is the first to describe the endodontic management of a maxillary first molar with a protostylid. CBCT has significant advantages in the clinical diagnosis. Owing to the aberrant morphology of the crown and the complexity of the root canal system, the treatment procedures for protostylid require special consideration.


Milia are common in children. They are small, keratinizing cysts that arise either as primary epidermoid cysts or secondary to other dermatoses, trauma or certain medications. In the paediatric population, milia are most frequently congenital and resolve spontaneously. Infantile hemangiomas are relatively common in neonates. They typically arise within the first few weeks of life, undergo a proliferative phase in the first 6 months, then begin to involute at around 12 months of age. After involution, residual skin changes may be observed, such as telangiectasia, fibrofatty tissue and redundant skin. However, there is a gap in the literature regarding concomitant milia and infantile hemangiomas. We report a case of a 5-months-old female who presented with a large, segmental infantile hemangioma of the posterior neck with milia. Primary milia arise from the sebaceous collar of vellus hair follicles and present as pearly, white-yellow papules. The most common type of primary milia is congenital milia, which are typically found on the face and present in 40%–50% of newborns without a sex predilection. Other causes of primary milia include certain genodermatoses, such as Bazex-Dupre-Christol syndrome. In comparison, secondary milia more commonly arise from eccrine ducts than from hair follicles and exhibit the same histopathologic findings as primary milia. Secondary milia are typically associated with other skin pathology, such as epidermolysis bullosa and other blistering dermatoses, as well as certain medications and skin trauma. Although milia are one of the most common, benign paediatric dermatoses, there is a paucity in data regarding their pathophysiology and significance in association with skin diseases.Milia are superficial, keratinizing epidermoid cysts that are common in children. Histologically, the walls of a milium are composed of stratified squamous epithelium with a layer of granular cells that encase keratin. Milia can be classified into two major categories: primary and secondary. It is hypothesized that the catalytic factor in IH development is transient hypoxia, which triggers increased angiogenesis in individuals with a genetic predisposition. Segmental hemangiomas are a subtype of IHs that cover an anatomic area, arise from one or more focal units, and measure ⩾ 5 cm. Cephalic segmental hemangiomas are more commonly associated with PHACES syndrome . Thus, prompt assessment and intervention are necessary.Infantile hemangiomas (IHs) occur in about 4.5% of neonates and are more common in Caucasian females. There have been no reported cases of milia arising in IHs. As such, the prevalence and significance, if any, of these comorbid conditions are unknown. Herein, we present an unusual case of a 5-months-old female who developed multiple milia overlying a segmental IH.Despite both milia and hemangiomas being relatively common dermatoses in children, there has only been one case report describing congenital hemangiomas with milia-like cysts.A 5-months-old female with Fitzpatrick I skin presented with four vascular tumours on the posterior neck that were first noted within the first few weeks of life. The tumours exhibited initial rapid growth over a 4-week period. They restricted the patient’s neck movement. She did not have any neurological symptoms, such as seizures or abnormal tone. She had an episode of apnea lasting 10 s in the first week of life and wheezing until she reached 3.6 kg in weight. The patient was delivered, weighing 2.5 kg, via emergency Caesarean section at 38 weeks due to failure of labour progression and small-for-gestational age. She is otherwise healthy and has met normal developmental milestones.On examination of the posterior neck, there was a band of four 4–5 cm tumours with overlying red-violaceous discolouration. On the right lateral nodule, there were multiple white papules 1–3 mm in size, consistent with milia . SuperioThe patient was started on propranolol 0.5 mg/kg/day and was titrated up to 2.5 mg/kg/day with follow-ups every 2 weeks. She has tolerated the treatment well with no reported side effects. Improvement in the size, colour and texture of all hemangiomas has been noted. On the most recent visit, the milia have completely resolved. One study described 3 × 4 cm congenital superficial hemangiomas with overlying milia-like cysts located on the scalp and back of a 4-months-old male. The authors theorized the finding to represent either a hamartomatous structure or be secondary to the hemangiomatous disruption of developing hair follicles. Histologically, they noted keratinizing cysts lined with stratified squamous epithelium and surrounded by immature hair follicles. Similar to our patient, the milia-like cysts spontaneously resolved as the hemangiomas gradually regressed. Furthermore, it is useful to differentiate milia from pustules, as reports of pustules in congenital hemangiomas have been documented and theorized to represent tissue necrosis secondary to rapid involution.Limited studies exist describing hemangiomas with milia. A recent cross-sectional study of 153 patients with segmental IHs did not identify secondary morphological features, such as milia.Due the relatively benign nature of both conditions, reporting bias may impact the prevalence of concomitant milia and hemangiomas in the literature. We hypothesize that the presence of milia represents a disruption of eccrine glands and/or hair follicles due to the growth and large size of the segmental IH. In addition, it is plausible that friction on posterior neck may have traumatized the skin, leading to milia. Interestingly, patient’s periumbilical IH did not have milia, which may support the skin trauma hypothesis. We hope the present case report may provide clinicians and patients with reassurance of this rare coexistence of these two common conditions.


Pediatric vascular anomalies are classified into vascular malformations and vascular tumors. While vascular malformations are generally anomalous vessels, vascular tumors arise from endothelial cells characterized by abnormal proliferation. Vascular tumors, also called hemangiomas, are subdivided into infantile and congenital hemangiomas. The differentiation of these anomalies can be challenging, and immunohistochemical staining is often employed for this purpose. The GLUT-1 (erythrocyte-type glucose transporter protein) stain is positive for the infantile type.Hemangiomas are usually found in the head and neck region. Their occurrence in the laryngeal region in infants tends to manifest in the subglottic region. Hemangiomas in the larynx mostly do not cause any symptoms until they are large enough to cause dyspnea, stridor, or hoarseness of voice. They are mostly treated in infants with propranolol or surgical excision.We report a case of an eight-day-old female infant who presented with a mass that recurrently protruded out of the mouth when she cried. The mass stopped protruding out of the mouth when the baby became restless, had respiratory distress, and refused feeds. Endoscopy of the pharynx and larynx showed a pedunculated hemorrhagic mass attached by a stalk to the left arytenoid. With cautery, the stalk of the lesion was severed from its attachment. The baby was discharged on the fourth postoperative day and histology reported a cavernous hemangioma.Seven months after the surgery, the baby is growing normally. Yearly follow-up endoscopies have been scheduled to evaluate for recurrence or residual disease. Pediatric vascular anomalies are classified into vascular malformations and vascular tumors. The vascular malformations generally comprise anomalous vessels, while the vascular tumors arise from the endothelial cells characterized by abnormal proliferation. The vascular malformations, though present at birth, grow slowly and mostly become enlarged due to hormonal activity (such as at menarche or during pregnancy) or in the setting of trauma or surgery. Vascular tumors, also called hemangiomas, are subdivided into infantile and congenital hemangiomas. The differentiation of these anomalies can be very difficult, and immunohistochemical staining is helpful for their differentiation. The GLUT-1 (erythrocyte-type glucose transporter protein) stain is positive for the infantile type. Infantile hemangiomas are generally not present at birth, and, if they do occur, they only appear as a reddened or pale area where the lesion subsequently appears. The congenital type, which involves high-flow vascular tumors, manifests at birth. The congenital type can involute over a period of a year or may persist .Hemangiomas are the most common tumors of infancy and are mostly found in the head and neck region . These lIn children, the presence of congenital laryngeal hemangiomas may cause stridor or dyspnea due to their subglottic location in most cases . The diaAn eight-day-old baby girl was referred to our tertiary healthcare facility, with a history of intermittent protrusion of a fleshy swelling out of the mouth since birth. The baby had a normal cry but was noted to have difficulty feeding when the swelling returned to the throat. The examination showed a pink, healthy baby with a reddish mass that periodically protruded out of the mouth when she cried, as shown in Video A tentative diagnosis of a pharyngeal mass was made with a differential diagnosis of a lingual thyroid gland. An initial ultrasound of the neck found a normal thyroid gland. A nasogastric tube was blindly passed with caution and tube-feeding was started since breastfeeding was difficult for the baby. During admission, the baby developed a fever, which was managed with IV cefazolin and IV paracetamol. While awaiting a CT scan report, the baby became restless and started crying excessively, and the mass no longer protruded from the oral cavity. Emergency examination of the pharynx and larynx under anesthesia was then done after successful endotracheal intubation.The intraoperative findings were as follows: i) a pedunculated reddish mass with a pedicle attached to the superior surface of the left arytenoid region with the mass completely buried in the esophagus, and ii) a normal oral cavity, pharynx, and laryngeal structures (apart from the left arytenoid).The reddish lesion was then teased out of the esophagus with forceps and the pedicle was severed with diathermy under direct laryngoscopy with a Chevalier Jackson laryngoscope. The specimen, i.e. the lesion as seen in Figure The baby recovered well after surgery and was discharged on the fourth postoperative day. The CT scan, which was received after the surgery, showed a poorly differentiated mass located in the left half of the nasopharynx, as shown in Figure Histology reported the following findings: sections show proliferation of thick and thin-sized blood vessels in the dermis of varying sizes admixed with lobules of fat. The overlying epithelium was ulcerated. Diagnosis: cavernous hemangioma.Seven months after surgery, the baby continues to grow very well.Vascular anomalies have been grouped into vascular malformations and vascular tumors. Vascular malformations are present at birth in most cases but grow slowly and are challenged to grow more by trauma and hormones. Vascular tumors, which result from endothelial hyperplasia, are classified into the infantile type and the congenital type. The infantile type is differentiated from all other vascular anomalies by the positive GLUT-1 staining , which, Laryngeal hemangiomas are mostly found in the subglottic region in infants with the adult type being found attached to supraglottic structures such as epiglottis, aryepiglottic folds, arytenoids, and false and true vocal cords ,8. In ouLaryngeal hemangiomas occur in different forms, i.e., cavernous, capillary, or mixed types. The capillary type tends to have larger vascular channels with deeper infiltration into the submucosa while the cavernous types are well-circumscribed and less infiltrative . Our casManagement of these lesions depends on their size. Small lesions may be treated with observation and follow-up. Larger lesions causing symptoms may require intralesional injections of corticosteroids, carbon dioxide laser or potassium-titanyl-phosphate laser, or surgical excision . GeneralCongenital laryngeal hemangioma may or may not present any significant clinical features. Babies born with masses in the oral cavity should undergo careful endoscopy to evaluate for such lesions and clinicians should maintain a high index of suspicion for laryngeal hemangioma. Surgical excision is an effective method of treatment for laryngeal hemangiomas causing respiratory distress or other obstructive symptoms.


Visceral venous aneurysms are exceedingly rare clinical entities reported in the literature. Venous aneurysms are usually acquired in origin, with most often portal hypertension as the underlying pathology. Most venous aneurysms are incidental findings on imaging. Complications of venous aneurysms like rupture with catastrophic outcomes had been reported. However, no clear guidelines exist regarding the management of portal venous aneurysms as most of the data is available only from case reports. Here, we report a rare finding of fusiform superior mesenteric vein (SMV) aneurysm with arteriovenous communication and portal vein thrombosis in the background of non-cirrhotic portal hypertension. Visceral venous aneurysms are rare clinical entities reported in the literature. With the portal venous system as the commonest site for visceral venous aneurysm, superior mesenteric vein (SMV) aneurysm accounts for approximately 9% of all visceral venous aneurysms only . Venous A 40-year-old female presented with complaints of abdominal pain in the periumbilical and right upper quadrant regions, which was dull aching in nature and moderate in intensity with no relation to food intake or any aggravating factors for three months duration. She did not have a history of fever, jaundice, gastrointestinal bleeding, or easy fatigability during this presentation. She underwent an open splenectomy for massive splenomegaly approximately 10 years back at another center. She also gave a history of variceal bleeding two years back which was managed with endoscopic variceal banding. She did not have any comorbidity and claims no addictions. On examination, she was thin-built with moderate abdominal distension due to ascites. Her blood investigations revealed a hemoglobin of 10.6 g/dl, a total leucocyte count of 14,660 cells/mm3, and a platelet count of 1,42,000 cells/mm3. Renal function and liver function parameters were within normal limits with serum bilirubin of 0.69 mg/dl, serum albumin of 3.7 g/dl, aspartate aminotransferase of 45 IU/L, alanine aminotransferase of 28 IU/L, and alkaline phosphatase (ALP) of 121 IU/L. Prothrombin time was 14.4 seconds with an international normalized ratio of 0.92. Upper gastrointestinal endoscopy revealed sclerosed esophageal varices with an inflamed fundus. Contrast-enhanced computed tomography (CECT) of the abdomen revealed a focal fusiform aneurysm of the proximal SMV measuring around 4 x 3.5 x 4 cm close to the portal confluence  Figure .The main portal vein is thrombosed and replaced by multiple periportal collaterals. The splenic vein was 5.5 mm at the confluence with patent splenoportal confluence. The liver was noted to have irregular margins with no focal lesions. Multiple peribiliary collaterals were noted with central intrahepatic biliary radicle dilation. Multiple mesenteric collaterals and splenic fossa collaterals were noted. Moderate ascites were noted. The symptomatic nature, size, and location of the aneurysm provided irrefutable evidence of the need for intervention. Based on imaging findings of the intense arterial phase enhancement of the aneurysm, with remote suspicion of SMA aneurysm, abdominal aortography was planned initially to further evaluate the aneurysm for possible endoluminal therapy. During abdominal aortography, a large aneurysmal filling in the late phase was noted following contrast injection into SMA, thereby confirming the presence of arteriovenous communication. Due to excessive tortuosity, an attempt to cannulate the aneurysm was futile, and the microcatheter could not be tracked. Therefore, after angioembolization of the aneurysm failed, surgical management of symptomatic aneurysm was considered. Intraoperatively, a nodular liver with massive ascites of approximately three liters was noted. A fusiform aneurysm of proximal SMV measuring 4 X 3 cm was noted. Proximally, SMV aneurysm was noted to be involved till just distal to the splenoportal confluence. Distal narrowing of the aneurysm is noted approximately 2 cm beyond the middle colic vein draining site of the SMV (Figure Dense collaterals were noted in the retroperitoneal region, along the root of the small bowel mesentery and peripancreatic region. The critical location of the SMV aneurysm with the proximal main portal vein thrombosis in the background of portal hypertension ruled out the option of aneurysmectomy. Therefore, an interposition mesocaval shunt was performed with an 8 mm dacron graft using a 6-0 Prolene suture material to minimize the perfusion pressure in the aneurysm, thereby reducing the chances of complications like rupture, thrombosis, and pain Figure .Her liver biopsy done intraoperatively was reported as non-cirrhotic portal fibrosis. Postoperatively, the patient received an injectable low molecular weight of heparin initially and later titrated with tablet acitrom after 48-72 hours. The patient was discharged after an uneventful postoperative course and on regular follow-up for six months with shunt patency confirmed with a Doppler ultrasound.In 1982, Schild et al. reported the first case report on an SMV aneurysm in a 60-year-old female during an evaluation for a cystic lesion near the head of the pancreas . Based oMost of the portal venous aneurysms were incidentally detected in asymptomatic patients . PatientTreatment of SMV aneurysms remains controversial with a spectrum of treatment options varying from close observation to endoluminal therapy and open surgical intervention. In a review by Sfyroeras et al., it was reported that portal venous system aneurysms do not require any form of treatment in most cases. In 88% of patients, who were followed up with abdominal ultrasound alone, the aneurysm diameter remained stable, and no complications occurred in any of the patients . In suppIn contrast to the conservative mode of the management of SMV aneurysms, guided only by the evidence of few observations, there has always been a call for surgical intervention even for asymptomatic SMV aneurysms. The proponents for this advocated that the prognosis for these patients with aneurysms is unpredictable and potentially described complications like rupture can be fatal . The letThe surgical intervention to be chosen depends on the location and shape of the aneurysm, the presence of complications, and associated comorbidities such as liver cirrhosis and portal hypertension. In patients with saccular aneurysms, aneurysmorrhaphy with or without venous patch angioplasty is the best and easiest surgical option preferred. In patients with fusiform aneurysms, aneurysmectomy with interposition bypass graft is the preferred approach. A bypass graft can be either an allograft from a cadaveric donor or a synthetic graft. In patients with documented portal hypertension and liver disease, the aim of surgical intervention is to decompress the portal system through a portosystemic shunt procedure and not to specifically treat the aneurysm. Portosystemic shunting is intended to prevent the ectatic vessel from rupturing and to reverse the dilatation by restoring the pressure in portal circulation to normal. The patient in the present report had the features of portal hypertension with ascites and proximal portal vein thrombosis with multiple collaterals in the periportal, retroperitoneal, and splenic fossa regions. Therefore, the patient underwent an interposition mesocaval shunt to decompress the portal venous system.In patients with predisposing conditions, any cystic lesion along the course of superior mesenteric vessels should be evaluated with caution for possible aneurysms. The location, shape, and presence of portal hypertension decide the choice of surgical option in patients with SMV aneurysms. With the minimal evidence available regarding SMV aneurysms and unpredictability in the onset of complications, the option of surgical intervention for SMV aneurysms even in asymptomatic good surgical risk patients cannot be ruled out.


This case report describes a patient who underwent laparoscopic resection of the mucocele of the appendix secondary to endometriosis, a rarity in clinical practice.The patient was a 38-year-old woman with a history of endometriosis and an ovarian cyst who sought medical advice with a chief complaint of mild right lower abdominal pain.Computed tomography and ultrasonography of the abdomen revealed a cystic lesion at the distal end of the appendix without definitive findings of malignancy. Colonoscopy revealed a submucosal tumor-like elevation at the appendiceal orifice. Appendiceal mucocele was suspected preoperatively.The lesion was resected laparoscopically. Secondary ileocecal resection with lymphadenectomy was possible if the resected specimen was pathologically diagnosed as a malignant tumor with the risk of lymph node metastasis.The resected specimen was pathologically diagnosed as an appendiceal mucocele secondary to endometriosis; therefore, additional surgery was avoided.Although appendiceal mucoceles secondary to endometriosis are rare, laparoscopic surgery in which only the lesion was resected is a useful strategy for the treatment and pathological diagnosis of appendiceal mucoceles without findings of malignancy. Neoplastic luminal strictures include benign and malignant tumors, and the rupture of these mucoceles sometimes produces pseudomyxoma peritonei. Endometriosis (0.05%–0.15% of appendectomies) is one of the rare causes of appendiceal mucocele because of nonneoplastic luminal strictures.,2,4 Although the mucocele of the appendix requires surgery, ileocecal resection with lymphadenectomy is often performed by laparotomy because of the concern for rupture and the risk of malignant tumors. Mucocele of the appendix secondary to endometriosis is rare, but we found that laparoscopic surgery for resecting only the lesion is a useful strategy for treatment and pathological diagnosis in cases without malignant findings. Our case report is the first to describe the pathological findings of an appendiceal mucocele caused by crypt gland obstruction resulting from fibrosis of endometriosis. Our study also describes laparoscopic surgery as a beneficial modality for treating mucocele of the appendix secondary to endometriosis.Mucocele of the appendix is a rare disease characterized by partial or complete distension of the appendix due to some obstruction or stenosis. This disease is observed in 0.08% to 0.33% of appendectomies.A 38-year-old woman with a history of endometriosis and an ovarian cyst sought medical advice with a chief complaint of right lower abdominal pain. On physical examination, the patient presented with no fever and mild right lower abdominal tenderness without rigidity. Blood tests revealed no evidence of inflammation. Computed tomography and ultrasonography of the abdomen revealed a cystic mass at the distal end of the appendix without definitive findings of malignancy Fig. A and B. We performed a partial laparoscopic resection of the cecum to resect the lesion completely Fig. A. We werThe resected specimen macroscopically showed the cystic lesion at the top of the appendix Fig. B. The cyThe postoperative course was uneventful, and the patient was discharged on postoperative day 4. Because endometriosis is a benign disease, additional surgery and follow-up were not required. After the surgery, the right lower abdominal pain disappeared.In this study, we described a rare case of appendiceal mucocele secondary to endometriosis. Because preoperative examinations and surgical findings did not reveal findings suggestive of malignant disease and rupture, we performed laparoscopic surgery to remove the lesion completely as a partial resection of the cecum. As a result, a rare condition of appendiceal mucocele secondary to endometriosis was diagnosed pathologically, and additional surgery was unnecessary. Some pathological entities, including benign and malignant diseases, cause appendiceal mucocele. Diagnosing benign or malignant appendiceal lesions preoperatively is challenging for anatomical reasons, except in cases with findings suggestive of malignant diseases such as metastasis or pseudomyxoma peritonei. Surgery is required to treat appendiceal mucocele, but the optimal surgical procedure differs depending on the causative disease. For benign diseases, resection of the lesion alone is sufficient. Although malignant, low-grade appendiceal mucinous neoplasms and T1a appendiceal cancer do not cause lymph node metastasis, thus resection of the lesion alone without lymphadenectomy can serve as sufficient treatment. Ileocecal resection with lymphadenectomy is required for more advanced appendiceal cancers than T1a. However, in clinical practice, if benign or malignant lesions are not diagnosed preoperatively, ileocecal resection with lymphadenectomy is often selected because of the risk of malignant diseases. This indicates that more invasive surgery has been performed in benign diseases or malignant tumors without the possibility of lymph node metastasis. Our study found that a pathological diagnosis using permanent specimens from the lesion resection can avoid unnecessary and invasive surgery.The appendiceal mucocele is macroscopic in appearance and presents as a cystic lesion where the appendix is partially or completely expanded.In our case, we could not make a preoperative histological diagnosis, but there were no findings suggestive of malignant disease, such as metastasis or pseudomyxoma peritonei. Therefore, we considered certain diseases, including appendiceal endometriosis, for which surgery can be performed by completely removing the lesion. Laparoscopic surgery is minimally invasive and esthetically superior and has the advantage of less adhesion when additional surgery is required. A pathological diagnosis of appendiceal endometriosis was made based on the laparoscopic surgery specimen, and excessive secondary surgery was unnecessary. Except for cases in which a malignant tumor could be diagnosed preoperatively, the first laparoscopic resection of the lesion alone is useful, with the option for a secondary surgery depending on the results of the histological diagnosis. A minority of appendiceal mucoceles are caused by retention of mucin secondary to obstruction or stenosis of the appendix due to fecaliths, post-inflammatory scarring, and rarely are they caused by endometriosis . Endometriosis is a common disease in women, defined as functioning endometrial tissue in a location outside of the uterus. Small cystic lesions called blueberry spots, in which menstrual blood accumulates, are often observed with endometriotic lesions, although there were no blueberry spots in our case. The pathological findings confirmed the relationship between the columnar epithelium and the endometriotic lesions using CD10, ER, and CDX2. Such histopathological diagnostic methods using immunostaining are useful in distinguishing endometriotic lesions from the columnar epithelium of the appendix and revealed findings of a rare mucocele of the appendix caused by a crypt gland obstruction of the appendiceal mucosa. Klingbeil et al reported a case where an appendiceal mucocele consisted of endometriosis and low-grade appendiceal mucinous neoplasms; therefore, our pathological findings may have had the potential of developing malignancy in the future.Although endometriosis of the appendix is rare, unique pathological findings were observed in our case. Most appendiceal mucoceles are caused by epithelial lesions associated with mucin production, such as hyperplastic polyps, adenomas, and carcinomas.,8–13 In these cases, endometriosis was the preoperative diagnosis in only 1 case in which endometriosis was diagnosed by previous laparoscopy. In these cases, appendiceal mucocele was caused by obstruction of the lumen of the appendix (5 cases), obstruction of the appendiceal lumen and the crypt gland of the mucosa (1 case), and appendiceal mucocele with an indeterminate cause (2 cases). Ileocecal resection was performed in 1 case, right hemicolectomy in 1 case with rupture, hysterectomy with appendectomy in 1 case with surgery for endometriosis, and lesion resection alone as appendectomy or partial resection of the cecum in 5 cases. Except for cases of rupture of the appendiceal mucocele and surgery for endometriosis, ileocecal resection was performed in 1 of 6 cases. None of the patients underwent laparoscopic surgery. To the best of our knowledge, our case report is the first to describe the pathological findings of an appendiceal mucocele caused by crypt gland obstruction resulting from fibrosis of endometriosis. Our study also describes laparoscopic surgery as a beneficial modality for treating mucocele of the appendix secondary to endometriosis.The obstructive mucocele of the appendix secondary to endometriosis is rare, and reported cases with a similar condition were previously described in only 8 cases.The case report has several limitations. En bloc resection cannot be achieved when 2 stages of extensive surgery are required in cases of malignancy. Therefore, if preoperative or intraoperative findings are positive for malignant tumors, such as metastasis or pseudomyxoma peritonei, ileocecal resection with lymphadenectomy should be selected. Because open surgery rather than laparoscopic surgery reduces the chances for intraoperative rupture of the mucocele of the appendix, careful surgical skills are required in laparoscopic surgery.Mucoceles of the appendix secondary to endometriosis are rare and difficult to diagnose preoperatively. In cases of mucocele of the appendix without definitive findings of malignancy, complete laparoscopic resection of only the lesion is a useful strategy for treatment and pathological diagnosis.www.editage.jp) for English language editing, and Ms. Satoko Fukaya who works at our hospital library.The authors thank Dr. Hiroko Ogata for her support in pathological diagnosis, Editage (Conceptualization: Hitoshi Hara.Data curation: Hitoshi Hara, Seito Shimizu, Yasuhide Muto, Tomoki Kido, Ryohei Miyata.Investigation: Hitoshi Hara.Resources: Hitoshi Hara, Ryohei Miyata.Validation: Hitoshi Hara.Visualization: Hitoshi Hara.Writing – original draft: Hitoshi Hara.Writing – review & editing: Hitoshi Hara, Ryohei Miyata.


Epithelioid hemangioendothelioma (EHE) is a rare condition arising from endothelial cells of the blood vessels. This is a vascular tumor that may occur anywhere throughout the body. This tumor behaves on a spectrum as either a benign tumor or an aggressive sarcoma. The EHE tumor and its management depend on the location of the lesion and accessibility for surgical excision. This case is a rare example of a patient presenting with a maxillary aggressive EHE tumor. This lesion was incidentally found on head CT done for ruling out fractures of the mid-face with incidental findings of an asymptomatic destructive lytic lesion. The treatment of this tumor located in a vital region of the mid-face will be discussed. Epithelioid hemangioendothelioma (EHE) is a rare vascular neoplasm with incidence of 0.038/100000 per year . The incThe presentation of EHE varies from asymptomatic and localized to systemic complications based on the area of involvement. Thus, EHE is often an incidental finding on CT or MRI. Possible symptoms of EHE include pain, mass effect, fever, or weight loss. At times, these patients can also present with venous obstruction in a specific organ or region leading to the diagnosis. Up to half of the cases of EHE arise from or are associated with a vein whereas, in the rest of the cases, recurring symptoms of edema or thrombophlebitis can occur due to venous occlusion facilitating the diagnosis of EHE. Additionally, undiagnosed anemia has been identified in multiple patients with EHE.Being a rare condition, consistent studies haven't been made to guide diagnosis and treatments. Suboptimal planning and treatments have affected patient outcomes and an attempt has been made to further study the best treatment options. Most findings have been associated with anecdotal cases, and no phase II or III trials have been conducted for EHE treatment. There are two ongoing prospective studies for the two medications: trametinib and eribulin ,7. ThereA 66-year-old female presented to the emergency room with abdominal pain. She was treated conservatively and sent home to follow up with her primary care physician. She experienced constipation while using the restroom and suffered a syncopal episode. This led to the loss of consciousness, a fall, and a head injury. She was subsequently transported to the emergency room for re-evaluation. En route, emergency medical technicians identified a systolic blood pressure in the 70s and administered intravenous fluids, Zofran, and checked her serum glucose levels. Her random serum glucose was 154 mg/dL at the scene.In the emergency room, she complained of mild right posterior parietal headache. She denied shortness of breath and any chest pain or discomfort. She also denied a history of blood in stool or bleeding history. Her medical history was significant for hypertension and asthma, and she had a history of pelvic mass excision three years ago which was benign. Her medications at the time of presentation included albuterol, calcium docusate, ferrous sulfate, losartan, and pantoprazole. She had a negative smoking history and an allergy to shrimp.Her blood pressure had stabilized at 125/59 with a normal heart rate, oxygen saturation on room air, and temperature. Her head, ears, eyes, nose, and throat (HEENT) exam was significant for tenderness over the right posterior parietal area of the scalp. Her right orbit was tender to palpation and she had some peri-orbital swelling. Her extraocular motions were intact and there was no entrapment of her orbital muscles. Abdominal and neurologic exams were unremarkable with normal bowel sounds. The rectal exam was negative for blood, and the stool appeared normal. Her chemistry was significant for sodium of 135 mmol/L, and her complete blood count (CBC) was notable for hemoglobin of 8 g/dL and hematocrit of 24.6. Chest X-ray was unremarkable. Left ventricular hypertrophy was noted on a 12-lead ECG.A head CT scan showed no acute abnormality and a probable left orbital floor fracture with blood in the maxillary sinus. A facial CT also had an incidental finding of a destructive lesion, which was concerning for osteomyelitis versus malignancy Figures , 2, 3. TWe were consulted for evaluation. She was taken to the operating room and a left Caldwell-Luc procedure with a biopsy of the intra-sinus mass was carried out. The mass was excised and debrided as much as surgical access allowed Figure . The resThe excision of the mass was done in combination with bone debridement. No margins could be obtained posteriorly and medially due to severe proximity to multiple vital structures including the posterior superior alveolar artery, inferior orbital artery, and branches of the trigeminal nerve. Once the patient was stabilized, she was discharged to be treated for this lesion after a full body workup and consideration for sequential imaging. A full body Scan was obtained with negative metastatic findings. On follow-up with oncology, the decision was made to observe the lesion with serial scans and if necessary in the future to provide localized radiation to minimize injury to adjacent structures. Epithelioid hemangioendothelioma is an extremely rare vascular condition affecting the epithelial cell lining of blood vessels. The tendency for this disease to behave as either a mild low-grade growing tumor to a severely aggressive disease. Presentations of EHE range from asymptomatic to severe pain  and malaise with the presentation of metastatic cancer. There have been cases of skin involvement which has been confused with other pathological entities. Epithelioid hemangioendothelioma is not a heritable condition, but some genetic changes have been associated with this condition. These include products of genetic translocations such as yes-associated protein1 (YAP1)-transcription factor E3 (TFE3) gene fusions or WW domain-containing transcription regulator 1 (WWTR1)-calmodulin binding transcription activator 1 (CAMTA1). There has been no consensus on treatment until the European Society for Medical Oncology (ESMO) in 2020 attempted to align patient treatments and provide guidelines for treatment and future research for EHE . Based oTreatment of EHE patients depends upon the location and spread of the disease. Surgery is the treatment of choice for localized accessible lesions. Excision with a small margin and following the tract of needle biopsy is the ideal treatment where a cure rate of 70% to 80% is expected with negative margins . In caseIn summary, the rarity of this disease has limited our understanding of the behavior of and treatment options suitable for EHE. More prospective studies are necessary to better understand and manage this condition. Case presentations like in our patient and follow-up of these rare patients help expand the understanding of these patients and their disease behavior. It is necessary to report these cases to increase the number of available data for the future care of these rare patients.This patient is a prime example of how these rare EHE patients present to healthcare providers. Due to the rarity of these cases, continuous case reports are necessary to give practitioners a broader view of their findings and disease behavior. This allows for improved outcomes for EHE patients in the future. The biopsy result is usually unexpected and a full metastatic workup including local, brain, and chest scans are necessary. In this patient, the unfavorable location of the lesion margin led to the decision of debridement, followed by close observation of the lesion. The lesion in this patient may have a benign behavior therefore in this patient, the radiation to vital structures was deferred until some progression and follow-up are done to see if her EHE would continue to grow or stop. At that point, radiation treatment would be considered.


Traumatic diaphragmatic injuries (TDIs) are rare and can be life-threatening, depending on the size of the injury and the contents herniating through it. They usually result from blunt or penetrating trauma to the thoracoabdominal area, with an incidence of 0.8-5% and up to 30% presenting late. A high index of suspicion should be maintained when evaluating patients with a history of trauma  and upper abdominal symptoms.We present a case of a missed TDI after a left posterior thoracoabdominal stab injury, which was evaluated with a diagnostic laparoscopy at an outside hospital. He presented to our emergency department (ED) with sudden onset left-sided chest pain and uncontrollable vomiting. A CT scan was obtained and showed a distended stomach herniating through a defect in the left hemidiaphragm. The patient was immediately taken for laparoscopic exploration and repair. There was a 5 cm defect in the left posterolateral diaphragm containing a strangulated stomach (approximately ⅔) and necrotic omentum. Complete reduction was achieved and the diaphragmatic defect was repaired primarily. His postoperative course was uncomplicated. This case illustrates the importance of maintaining a high index of suspicion for TDI, despite reports of previous exploration. Missed TDI can present with herniated intra-abdominal organs, which can become strangulated and increase morbidity and mortality. Traumatic diaphragmatic injuries (TDIs) are rare, with only a 0.8-5% incidence rate, but up to 30% of cases present late. These injuries can be life-threatening, depending on the size of the rupture and contents that herniate through it, as well as the presence of strangulation or obstruction . TypicalWe present a case of a 31-year-old Hispanic male with a one-year history of a left posterior thoracoabdominal stab injury, evaluated with a diagnostic laparoscopy at an outside hospital. He presented to our ED with sudden-onset left-sided chest pain and uncontrollable vomiting. He endorsed chills but no fever. A chest X-ray (CXR) was obtained and reported as unremarkable at that time; however, showed a suspicious elevation in the left hemidiaphragm compared to a previous CXR six months prior Figure . GeneralDue to the systemic and laboratory signs of developing ischemia in the setting of a herniated stomach with a risk of stomach perforation, the patient was taken for laparoscopic abdominal exploration. There was a 5 cm defect in the left posterolateral diaphragm Figure  containiHis postoperative course was uneventful, he was extubated on postoperative day one, and on postoperative day five, the chest tube was removed after the output was <10 ml in 24 hours for the past 48 hours. He was discharged from the hospital on postoperative day five. During his follow-up visit three weeks after the surgery, he had no issues, and further appointments were scheduled as needed.TDIs are uncommon and can result from both penetrating and blunt trauma. The reported incidence ranges from 0.8% to 8%, but it is challenging to determine the actual number due to the injuries that may not present until days or even years later, with rupture of the diaphragm and subsequent herniation of abdominal contents . A studyA high degree of suspicion is required for proper diagnosis of these injuries as they are often missed even after exploratory laparotomy or laparoscopy. The latter is likely due to the difficulty with direct visualization of both hemi-diaphragms . Also, tDelayed presentation is the single most important contributor to increased mortality . This unDiaphragmatic injuries may only be detected after herniation of abdominal contents occurs. The most commonly herniated organs are the stomach, spleen, and bowel. Left-sided ruptures are more common but right-sided ruptures are more difficult to diagnose. The preponderance of left-sided injuries is likely related to the protective effect of the liver on the right side .Diagnosis represents a challenge for clinicians, and in many cases, the patient’s chief complaints are nonspecific, such as chest pain, vomiting, nausea, abdominal pain, general malaise, and breathlessness . LiteratOnce the diagnosis of diaphragmatic rupture has been established, management is with surgical reduction of the herniated contents and defect repair. Conservative treatment is not advised as complications involving volvulus, obstruction, or strangulation can arise . SurgicaSome of the benefits of the laparoscopic approach include less postoperative pain, faster recovery, and fewer wound complications . The useIt is important to note that TDIs have an overall mortality rate of 21% . TherefoTDIs with herniation of abdominal contents are a rare entity; however, they cause significant morbidity and an overall mortality of 21%, or worse when diagnosis is delayed and/or missed. One of the most severe consequences is strangulation followed by sepsis, as was the case with our patient whose systemic and laboratory signs indicated ischemia, later confirmed by intraoperative findings. Therefore, it is crucial for trauma centers to improve their understanding of these injuries to prevent delayed diagnosis. A high index of suspicion should be maintained to potentially diagnose diaphragmatic injuries in patients with the mechanism for a potential injury. If a TDI is diagnosed and repaired on initial presentation, this could avoid later organ herniation through the defect. However, when evaluating patients with a history of blunt or penetrating trauma in the thoracoabdominal region presenting with upper abdominal symptoms, it is key to suspect a late presentation of TDI with herniation of abdominal contents.