Background: Retrocaval ureter is a rare entity with a reported incidence of ∼1 in 1100 and a 2.8-fold male predominance. The course of the ureter could be classified, using an intravenous urography, as type 1 having S-shaped, fish-hook, or J-shaped retrocaval course or type 2 having sickle-shaped course. No case report describing retrograde endoscopic management of ureteral calculi in the presence of retrocaval ureter could be found in existing literature. We are presenting a case of type I retrocaval ureter with ureteral calculi and nonobstructive drainage, which was effectively managed by flexible ureteroscopy.Case Presentation: A 62-year-old Caucasian man presented with complaints of a renal colic. The patient was positive for a history of noninsulin-dependent diabetes and hypertension. A direct abdomen CT scan showed an 8 mm ureteral stone with suspected retrocaval course of right proximal ureter with no hydronephrosis. After informed consent, ureteroscopy was performed on the patient's right proximal ureter. No complications occurred intraoperatively and postoperatively. On follow-up of up to 3 months, patient was asymptomatic and direct abdomen CT scan showed normal kidney without hydronephrosis.Conclusion: In the presence of retrocaval ureter and associated ureteral calculi with a condition of nonobstructive drainage, retrograde ureteroscopy is a safe and optimal procedure. Using an intravenous urography (IVU), the course of the ureter could be classified as type 1 having an S-shaped, fish-hook, or J-shaped retrocaval course or type 2 having sickle-shaped course. Symptoms caused by the obstruction typically appear in the third or fourth decade of life. A surgical treatment is necessary in these cases and it consists of a mobilization of the ureter both above and below the retrocaval course and of an ureteroureterostomy. Sometimes, this vascular anomaly is not always associated with ureteral obstruction. The management of a patient with renal calculi along with a retrocaval ureter is hence laden with a dilemma: treat the renal calculus alone or also repair the ureteral anomaly. No cases report describing retrograde endoscopic management of ureteral calculi in the presence of retrocaval ureter could be found in existing literature. We are presenting a case of type I retrocaval ureter with ureteral calculi and nonobstructive drainage, which was effectively managed by flexible ureteroscopy.Retrocaval ureter is a rare entity with a reported incidence of ∼1 in 1100 and a 2.8-fold male predominance. The first observed case of retrocaval ureters was described by Hochstetter in 1893.2)).A 62-year-old Caucasian man presented with complaints of a renal colic. The patient was positive for a history of noninsulin-dependent diabetes and hypertension. History of fever, hematuria and dysuria, and loss of weight were absent. Clinical examination of the abdomen was within normal limits. Complete laboratory evaluation, including urinalysis, complete blood picture, urea, creatinine, and electrolytes, showed a mild renal insufficiency  and without a ureteral sheath, to avoid an accidental perforation of the ureter at the retrocaval curve, a ureteroscopy was performed. The stone during the previous maneuvers was pushed up in the kidney. The stone was easily found in an inferior calix . The anomaly predominantly involves the right ureter.Retrocaval ureter may be asymptomatic or discovered during radiological imaging for some other problems. However, some time during the excretory phase of CT scan entire ureter is not viewed because of pooling of contrast in the dilated renal pelvis and proximal ureter and this problem also occur in our case. Although not diagnostic, the appearance of retrocaval ureter on IVU is typical and is highly suggestive of the diagnosis. If the patient is symptomatic with documented subrenal functional obstruction, dismembered pyeloplasty is the gold standard treatment.Surgical management is reserved for type 1 cases that are usually symptomatic. Patients with minimal caliceal dilation and without significant symptoms do not require surgery, but they need to be followed up.2 have reported simultaneous treatment of renal stone and retrocaval ureter with laparoscopic technique.Various reports are present in literature describing association of retrocaval ureter and renal calculi with their simultaneous surgical management. Simforoosh et al.3 The author was of the opinion that the placement of a Double-J stent probably altered the course of the ureter from long J shape to a less curved sickle shape, which offered little hindrance in clearance of stone fragments.No case reports describing management of renal calculi by ureteroscopy and its feasibility in the presence of retrocaval ureter could be found in existing literature. A case report described the feasibility of extracorporeal shock wave lithotripsy in the management of renal calculus in patients with retrocaval ureter4A case of an effective percutaneous nephrolithotomy treatment of a kidney stone in a type I retrocaval ureter was described by Prakash et al. in 2013 with effective results.Our patient had type I retrocaval ureter with concomitant ureteral calculi. Considering the nonobstructive drainage of the kidney, we treated only calculi.In the presence of retrocaval ureter and associated ureteral calculi with a condition of nonobstructive drainage, retrograde ureteroscopy is a safe and optimal procedure.


An effective long‐term treatment is necessary for mares with pyometra, because the condition tends to recur. In many affected animals, several conformational or anatomical anomalies contribute to impaired uterine clearance. Ovariohysterectomy is the surgical procedure of choice. Conservative therapy consists of draining and flushing the uterus, and systemic anti‐inflammatory and antimicrobial treatment. Uterine secretions tend to accumulate again after local treatment, especially in mares with poor vaginal conformation or cervical adhesions. Herein, we describe three cases in which a cervical stent was used in mares after mechanical or manual dilation of the cervix to achieve permanent draining of the uterus. The mares remained symptom‐free for up to 6 years and exhibited good clinical progress and good performance in competitions. Potential complications of the procedure include loss of the stent and obstruction caused by viscous secretion. A cervical stent is a relatively easy and low‐cost option for the long‐term treatment of pyometra in mares, particularly in cases where excessive costs of surgery and risks of a general anaesthesia are to be avoided. The mare had given birth to 1 foal several years ago without complications. The vagina and cervix exhibited massive adhesions. Manual passage of the cervical canal was possible, and the uterus was flushed with saline. The mare was treated with antibiotics and non‐steroidal anti‐inflammatory drugs for 7 days. Over the next 18 months, the mare regularly exhibited symptoms of colic and fever, and there was constant accumulation of fluid in the uterine lumen. In February 2012, a permanent catheter (stent) was placed in the cervix under sedation and epidural anaesthesia. Beforehand, the cervical canal was dilated with a balloon used for catheter interventions in small animal surgery. The stent was manufactured by G. Herber, a technician of the faculty of veterinary medicine according to the authors' requirements. It consisted of a flexible connecting hose (length 10 cm) used for sanitary installations combined with a plate and screw nuts at both ends  indicated that she had not been pregnant for more than 35 days.An 18‐year‐old warmblood mare was presented with colic and fever 8 weeks after her last insemination. She had given birth to one foal several years previously, had failed to become pregnant earlier in that season, but was diagnosed as being pregnant with large amounts of foetal fluid 2 weeks before presentation. To support luteal function, she had been administered the gestagen altrenogest over the last 4 weeks. Due to the colic, the mare had been treated with anti‐inflammatories and antibiotics, without success. During a gynaecological examination, 35 L of purulent secretion was removed from the uterus. Passage through the cervix was easy, but palpation revealed a solid consistency presumably due to fibrosis. Flushing of the uterus, oxytocin injections and antimicrobial therapy were carried out. Bacteriological culture from the uterus revealed moderate amounts of As the mare exhibited recrudescent filling of the uterus with purulent secretion during the following weeks, a cervical stent was administered 3 weeks later. To avoid corrosion, the stent was covered by a shrinking tube, as a modification to the stent used in case 1. The catheter had to be replaced with a system with a larger caudal plate . Resection of the enlarged and fluid‐filled uterus can be difficult, and the risk of peritonitis or wound infection is increased due to potential contamination of the abdomen. Furthermore, the complete removal of the uterus is impossible and remaining uterine tissue puts the mare at a high risk to develop postoperative complications. Thus, this surgery is a less common treatment in horses than it is in other species such as dogs , stent implantation can be difficult, but in the majority of mares, manual dilation of the cervix under mild sedation, and if necessary epidural anaesthesia, is adequate.Adverse side effects and risks associated with the procedure are marginal. Loss of function can occur if the stent is lost, shifts in the cervical canal or is blocked by detritus. If necessary, the lumen can be cleared by careful flushing with saline at regular intervals to dilute viscous secretions. In the follow‐up examinations of the above‐described cases, none of the mares exhibited blockage of the stent, but they had exhibited regular vaginal discharge. Loss of the first stent occurred in case 2 very soon after implantation because that stent did not fit tightly enough into the mare's dilated cervical canal. Adjustment of the pieces of the stent resulted in a secure fit in the cervix. A larger piece at the caudal part prevents movement into the uterine lumen. The stent was lost several years after implantation in case 1, and before this occurred, it had not been re‐evaluated for several years. Otherwise, the displacement of the catheter could have been detected and corrected earlier. Nevertheless, the asymptomatic interval was 6 years. The persistency of vaginal discharge after loss of the stent suggests that during the long time in place, the stent leads to structural changes in the fibrotic cervix which now prevents complete closure.In conclusion, pyometra in mares is a devastating disease in terms of breeding capacity but adequate treatment can result in a good condition without clinical symptoms and good general performance.None of the authors have any conflict of interest to declare.J. Krohn: examined and treated all mares, developed the modification of the stent and wrote and revised the manuscript. S. Ennen: investigated the mare presented in case one and was involved into in invention and implantation of the first stent. She revised the manuscript. R. Hospes: was involved into invention and implantation of the first stent and folllow up examinations in case one. He revised the manuscript. J. Nieth: examined and treated the mare presented in case three. She was involved into implantation and modification of the stent in case three and revised the manuscript. A. Wehrend: supervised examination and treatment of all presented cases and revised the manuscript.


Other than adipocytes, lipomas may contain mesodermal components such as varying proportions of fibrous tissues and blood vessels. Fibrolipoma is an uncommon variant of lipoma and comprises a high proportion of fibrous components. An intrathoracic fibrolipoma is extremely rare; to the best of our knowledge, only three such cases have been reported till date.A 51-year-old female presented with a left intrathoracic mass, which was confirmed to be a lipomatous tumor using computed tomography. A pedunculated tumor originating from the parietal pleura was resected using complete thoracoscopic surgery. Pathological examination indicated a diagnosis of fibrolipoma. Intrathoracic fibrolipomas are extremely rare; this is one of the first reported cases of successfully resecting an intrathoracic fibrolipoma using complete thoracoscopic surgery.The tumor was asymptomatic and relatively small when detected during a medical checkup. This enabled the successful resection of the tumor via complete thoracoscopic surgery. Although fibrolipomas are histologically benign, careful observation and follow-up are essential owing to the possibility of recurrence. Lipomas may contain mesodermal components other than adipocytes, including varying proportions of fibrous tissues and blood vessels. Fibrolipomas are an uncommon variant of lipomas that contain a high proportion of fibrous components. Fibrolipomas are reported to develop during the maturation of lipoblastomatosis; the maturation of adipose and fibrous tissues causes collagen fibers to separate fat cells into lobules, which is a characteristic feature of fibrolipoma –3.Lipomas account for 20% of all benign soft-tissue tumors; however, fibrolipomas are rare and account for only 0.03% of all benign soft-tissue tumors . They caIntrathoracic fibrolipomas are extremely rare. To the best of our knowledge, only three such cases have been reported till date , 2, 5. HA 51-year-old female with no subjective symptoms was referred to our hospital with a left intrathoracic mass that was discovered during a medical checkup. Computed tomography (CT) revealed a neoplastic tumor (43 × 48 × 13 mm) with extrapleural signs, located in the dorsal portion of the left thoracic cavity , particularly with fat saturation, is helpful for assessing the lipomatous nature of the tumor. In addition, MRI aids in distinguishing between lipomas and well-differentiated liposarcomas based on margins, signal homogeneity, and septa or nodules . PET imaIn the present case, intraoperative, frozen-section analysis revealed that the tumor was rich in fibrous components and demonstrated no malignancy. Generally, benign lipomas and liposarcomas can be distinguished depending on the presence of hyperchromatic nuclei; however, the identification of atypical stromal cells is not always easy. Thus, establishing a differential diagnosis using only intraoperative frozen-section analysis is difficult for pathologists. Because the differentiation of lipomas and liposarcomas is essential for proper patient management, it is important to conduct a detailed examination of the whole resected specimen.The lipomatous tumors exhibit varied clinical course. The first-line treatment for these tumors involves surgical resection. The prognosis is favorable for benign lipomas and well-differentiated liposarcomas after complete resection. However, high-grade tumors such as pleomorphic liposarcomas may develop distant metastases.Fibrolipomas present clinical characteristics similar to those of lipomas. Intrathoracic fibrolipomas are usually asymptomatic; however, the development of tumor causes the compression of surrounding organs, resulting in a shortness of breath and dysphagia . In the In the present case, the tumor was asymptomatic and was relatively small upon when first detected. Therefore, successfully resecting the tumor using complete thoracoscopic surgery was possible. Although fibrolipomas are histologically benign, careful observation and follow-up are essential owing to the possibility of recurrence .


Lipoma is a benign neoplasm that primarily affects the middle‐aged individuals and has a rare oral cavity occurrence. Given its noninvasive behavior and low recurrence rate, surgical conservative management should be regarded as the best therapeutic option. This paper highlights two patients along with their improved conditions following the treatment. Lipoma is found more often in the buccal mucosa, which is full of adipose tissue owing to adjacency to the buccal fat pad.1.2The etiology of oral lipoma is unclear. Some studies have acknowledged that mechanical factors, endocrine system, inflammation, obesity, chromosomal abnormalities, radiation, trauma, mucosal infections, and chronic irritation can contribute to the development of oral lipoma.1.3The main findings of the histopathological view of both soft tissue and intraosseous lipomas are arrangements of mature adipocytes that are divided into lobules by the connective tissue septae. Usually, a thin fibrous capsule surrounds the tumor. Several types of soft tissue lipoma are described based on microscopic variations. The most common type is fibrolipoma, which is characterized by the presence of the fibrous components adjacent to the fat cells. Other types such as osteolipoma, chondrolipoma, intramuscular or infiltrating lipomas, salivary gland lipomas, pleomorphic lipomas, angiolipomas, myxoid lipomas, spindle cell lipomas, and atypical lipomas are scarce.1.4Most lipoma cases are adult patients aged 40‐60 years. These tumors are slow‐growing, painless, soft, circumscribed, and associated with submucosal nodules with either a sessile or a pedunculated base.Most cases of IOL are accidentally diagnosed during a radiographic examination. The symptoms of IOL are different depending on its size, position, evolution, and growth rate. This tumor may be associated with pain, swelling, and numbness.Adequate surgical excision without a safe margin, which has a rare recurrence, is the treatment of choice for this tumor.22.1A 33‐year‐old woman referred to an orthodontist due to mandibular anterior crowding. While assessing her panoramic radiography Figure , her den2.2A 25‐year‐old woman referred to a maxillofacial department. Her chief complaint was painless swelling in the buccal mucosa for about 2 years, which interfered with her dental occlusion. The lesion was about 1.5 cm and mainly soft on palpation Figure . The les3The present study reviewed the literature of the past 10 years on oral and intraosseous lipomas in the PubMed database (Table In the present review, the authors presented two cases of oral lipoma.4A large number of articles were found, from which 77 articles were selected after applying the selection criteria Table . Among tBuccal mucosa was the most common region for the occurrence of oral soft tissue lipoma, and mandibular body was the most common site for oral intraosseous lipoma. Simple lipoma was the most common histopathological pattern in oral soft tissue lipoma. Most authors considered surgical technique as a definitive treatment.5Lipoma is a benign tumor that can occur in any part of the body. Lipoma can be found in both soft and bony tissues.The clinical features of intraoral lipoma can be related to the location of the lesion. They often refer to slow‐growing tumors associated with fatty tissue and vary in diameter, which contributes to the possibility of misdiagnosis.There are different reports about the relationship between lipoma and sex. The incidence of oral lipoma has been reported to be identical in the males and females, or male prevalence has been emphasized or vice versa.Bone lesions are often discovered by accident. Radiographic images show unilocular or multilocular radiolucent lesions with a honeycomb or soap bubble appearance and often an osteosclerotic border.The differential diagnosis of IOL includes simple cyst, post‐traumatic cyst, aneurysmal bone cyst, giant cell granuloma, ameloblastoma, osteoblastoma, arteriovenous malformations, hemangiomas, infarcted bone, chondrosarcoma, and liposarcoma.Complete surgical excision is the main treatment of lipoma. There is no recurrence after adequate excision. Injectable steroids are used to manage soft tissue lipoma, which can cause the atrophy of the adipose tissue and reduce the size of the tumor. Monthly injection of 1:1 mixture of lidocaine and triamcinolone acetonide is recommended to be administered to the center of the lesion.None declared.ND, FR, TP and XM: involved in the conception and design of the work, data collection, drafting of the manuscript, critical revision of the manuscript, and final approval of the version to be published.


Intrapulmonary bronchogenic cysts are rare in adults and most are outside the trachea and bronchi. There are reports of clear link with the trachea, but the cyst itself occurs outside the trachea. Thus, bronchoscopy will not reveal the cause, which often leads to a diagnosis by surgical resection. We herein report an extremely rare case of an intrapulmonary bronchogenic cyst that was located entirely within the left main bronchial lumen. Bronchoscopy revealed a shiny and smooth surface mass with abundant blood vessels in the lumen that blocked the left main bronchus. The patient was successfully treated with bronchoscopic resection and remained stable at 16 months of follow‐up. To our knowledge, this is the first reported case of an intrapulmonary bronchogenic cyst located entirely within the bronchial lumen. Polypoid bronchogenic cysts are rare congenital bronchopulmonary malformations, commonly occurring in the mediastinum or occasionally in the intrapulmonary region. Intrapulmonary bronchogenic cysts are usually found in the lower lobes The patient was a 70‐year‐old man who presented with exertional dyspnea, which had persisted for one week and aggravated dyspnea, which had persisted for two weeks. He had been diagnosed with asthma two years previously and had regularly used inhaled corticosteroids since then. He had a 48 pack‐year smoking history. On admission the patient had decreased breath sounds and coarse crackles in the left lower lung field. A laboratory test showed a normal blood leukocyte count and abnormal partial pressure of oxygen (59%) and oxygen saturation (88%) values. Chest radiography revealed an area of high density in the left lower lung, which led to a diagnosis of obstructive pneumonia and atelectasis Fig. A. ComputFlexible bronchoscopy revealed a shiny mass with a smooth surface and abundant blood vessels blocking the left main bronchus Fig. A. When gIntrapulmonary bronchial cysts, which account for 12–23% of reported bronchogenic cysts, are rare The clinical symptoms of bronchogenic cysts are diverse and are related to their size and the degree of organ compression. When the cyst is located entirely within the main bronchial lumen, it is easy for the severity of the symptoms to increase. In the present case, the cyst grew in size and blocked the large airways, causing symptoms similar to bronchial asthma, but without infection, which is different from ordinary cases.MRI is very useful for the diagnosis of bronchial cysts. They are characterized by a very high and uniform signal intensity on T2‐weighted imaging In conclusion, intrapulmonary bronchogenic cysts may occur in the bronchial lumen. Bronchoscopic treatment may be effective in such cases.Appropriate written informed consent was obtained for publication of this case report and accompanying images.


Malakoplakia is a rare granulomatous disease of the genitourinary system. Gastrointestinal tract is the second most common site of involvement. It usually mimics a malignancy but its association with adenocarcinoma has been rarely reported. A 59-year-old male patient with the history of weight loss and rectal bleeding for two months prior to administration was referred to our hospital. Pre-operative CT scan revealed a large sigmoid colon mass with the extension and invasion to the serosal surface as well as multiple regional metastatic lymph nodes. The patient underwent sigmoidectomy with the primary pathologic diagnosis of adenocarcinoma. Pathologic examination revealed a moderately differentiated adenocarcinoma invading peri-colic adipose tissue and inflammatory reaction compatible with malakoplakia at the invasive borders of the tumor with the extension to the serosal surface.In the patients with gastrointestinal malakoplakia, the presence of possible adjacent malignancy should be screened. The possibility of over-staging should also be considered for adenocarcinoma cases in association with malakoplakia. Malakoplakia is a granulomatous disease of infectious origin . This raA 59-year-old male patient with the history of rectal bleeding, weight reduction (10 kg within two months) and altered bowel habitus was referred to our hospital. The past medical, drug, and family histories were negative. On the physical examination, the abdomen was normal without any organomegaly. The laboratory study revealed anemia and high serum carcinoembryonic antigen (CEA) of 786 ng/ml. The liver and renal function tests were normal. On colonoscopy, a large mass with the size of 22 to 29 cm was identified from anal verge along with two small polyps located at 10 and 15 cm from anal verge. The computed tomography (CT) scan showed a sigmoid mass with the extension to the serosal surface and involvement of multiple regional lymph nodes without distant metastasis. The pathologic diagnosis was adenocarcinoma, therefore, sigmoid and upper rectal portions were resected with the stapled end to end anastomosis. On the pathologic macroscopic assessment, polypoid sigmoid mass surrounded by seven pedunculated and sessile polyps were identified. Macroscopically, the tumor was extended to the visceral peritoneum, which was suggestive for the pathologic stage T4a. On the microscopic examination, moderately differentiated adenocarcinoma, surrounded by adenomatous and hyperplastic polyps were noted. The tumor invaded into the pericolic adipose tissue through the muscularis propria. Infiltration of the sheets of foamy histiocytes with granular cytoplasm containing concentric basophilic inclusions, which were positive on Periodic Acid Schiff (PAS), Perl’s and Van-kossa stains (Michaelis-Gutman bodies), were observed between and beyond the tumor nests ,2. This Malakoplakia is a rare chronic inflammatory process that usually involves genitourinary organs. Gastrointestinal tract is the second most common site of involvement with predominant involvement of colon and rectum . MalakopE.coli and unusual stromal response to the carcinoma are the other possible hypotheses. The suggested pathophysiology of malakoplakia includes unusual inflammatory response to the altered normal flora in association with colon cancer, abnormal immune response and defect in the macrophage-lysosomal function. Infection with some bacteria including Although malakoplakia usually mimics malignancy, less than 30 cases of malakoplakia in association with adenocarcinoma of colon and rectum have been reported in English literature so far -12. PrevJadhav et al  reportedDespite the possibility for errors in clinical or intra-operative over-staging of colorectal adenocarcinoma in association with malakoplakia, the treatment planning and prognosis would not be altered.In the patients with gastrointestinal malakoplakia, the presence of possible adjacent malignancy should be screened. The possibility of pre-surgical over-staging for adenocarcinoma in association with malakoplakia must be considered as well.


Plexiform fibromyxoma is a rare and newly described gastric mesenchymal tumor with only 121 reported cases in the literature. Our understanding of plexiform fibromyxoma requires updating since the first case has been reported by Takahashi et al. 12 years ago. The present review summarized reported cases in the literature, and both clinical and pathological aspects of plexiform fibromyxoma were comprehensively discussed. Plexiform fibromyxoma usually causes nonspecific or bleeding signs or symptoms, and therefore clinical recognition of the disease is challenging. Plexiform fibromyxoma is of benign nature without any metastasis or recurrence reported, and more conservative surgical treatment should be considered. Plexiform fibromyxoma (PF), also known as plexiform angiomyxoid myofibroblastic tumor (PAMT), was first described in 2007 by Takahashi et al. This rare tumor of mesenchymal origin is typically seen in the stomach and shows clinically benign behavior. As implied by the name, it features a plexiform cellular architecture of a myofibroblastic nature, encircled by a myxoid intercellular matrix with rich vasculature .To date, only 121 cases have been reported in the literature with a designation of PF or PAMT; the clinical features of reported cases are summarized in Takahashi et al., in 2007, reported 2 cases of gastric plexiform fibromyxoma using the term “plexiform angiomyxoid myofibroblastic tumor (PAMT)” because the condition was morphologically distinct from other gastrointestinal mesenchymal tumors due to its bland spindle cells in a plexiform pattern, myxoid intercellular matrix with hypervascularity, and myofibroblastic nature . In 2008In 2009, Miettinen et al. described 12 cases of the disease and designated the tumors as “plexiform fibromyxoma” simply due to their cellular architecture and fibromyxoid nature . They alSing et al. proposed that PF and PAMT are two related, but different, entities at two respective fibroblastic and myofibroblastic ends of a spectrum . Yet, duSince the WHO classification had designated the nomenclature PF for this entity, to avoid confusion in the literature, we suggest the use of PF in consideration of epidemiological surveillance and scientific communication purposes. The term PAMT literally describes the features of the disease in more detail, but PF could be viewed sensu lato as a broader nomenclature that covers the disease as well as other variants. We consider this to be acceptable, given that the disease is a spectrum with variations. The final designation or subsets under PF could be further categorized in the future after consideration of sufficient case numbers and advanced investigations.According to the 110 cases reported in the literature from 2007 to 2018, the frequency of PF is more than 9.17 cases per year worldwide. However, we feel this is an underestimate, because clinical recognition of the disease has only been increasing since 2007, and some cases are assumed to have been misidentified as gastrointestinal stromal tumors (GISTs) or other entities. Miettinen et al. estimated that the frequency of PF is 150 times lower than gastric GIST . TherefoThe patient ages showed a broad range, from 5 to 81 years . Most patients were middle-aged, with a peak around 30–60 years old. This age distribution of PF was in approximate accordance with previous reviews , 51; howThe races of patients were not provided in most of the studies; consequently, the countries of the studies were substituted for the ethnicity of the patients unless the ethnic information of the patients was given. In terms of regions, most cases were reported from East Asia , followed by North America , Europe , South-East Asia , South Asia , and Africa . In terms of countries, most cases were reported from China , followed by the United States , Belgium , Japan , and Korea . This distribution may not reflect the genuine epidemiological status of PF, since a higher frequency may result from a higher quality of healthcare and from a larger population. Nevertheless, it still suggests that PF is a worldwide disease with a predominance in East Asia.PF has a benign nature but is associated with hypervascularity; therefore, the clinical presentation can range from incidental findings to nonspecific gastrointestinal (GI) symptoms and further to severe GI hemorrhage. The typical presentation of PF is a nonspecific gastrointestinal complaint, such as abdominal pain, abdominal distension, and abdominal discomfort. Hemorrhagic gastrointestinal presentations are also commonly seen, with consequent GI bleeding-associated presentations like anemia, melena, and hematemesis , 68, 74.Although initially categorized as a gastrointestinal mesenchymal tumor , PF has The size of the tumors, available in 98 cases, ranged from 0.8 to 17 cm in the maximal diameter, with an average size of 4.81±3.30 cm and a median size of 4.0 cm. Endoscopic visualization reveals that PFs are typically pink or reddish and glistening tumors, elastic in texture, and covered with ulcerative, erosive, or smooth mucosa. Endoscopic ultrasonography indicates that PFs are hypoechoic with mild heterogenicity. Macroscopic examination shows a classical PF appearance as a lobulated tan-white or grayish-whitish mass, gelatinous on the cut surface, cystic, with mucinous fluids, a multinodular or polypoidal growth pattern, unencapsulated, and with well-defined (but sometimes ill-defined) margins. Hemorrhage is commonly encountered. PFs mostly originate from the submucosa and muscularis propria, with extension ranging from the mucosa to the serosa, causing ulcer and/or perforation.p<0.0001). The difference in tumor size between ulcerative or erosive lesions and nonulcerative lesions was not statistically significant, as calculated by an independent Student's t-test (p=0.184).The condition of the tumor surface was reported in 76 cases: 50 (65.8%) were ulcerated, 22 (28.9%) were nonulcerated, and 4 (5.3%) were covered with eroded mucosa. Ulceration or erosion of the tumor was significantly associated with hemorrhage-related signs or symptoms, as determined by Pearson's chi-squared test , SMA (89.1%), and MSA (90%); however, these are nonspecific markers for mesenchymal and myofibroblastic lineages and were therefore sensitive but specific for diagnosing PF. Equivocal staining results were demonstrated for desmin, caldesmon, calponin, CD10, estrogen receptor (ER), and progesterone receptor (PR). Desmin and caldesmon are more specific markers for muscular lineage toward terminal muscle cell differentiation and exhibited limited and focal reactive results in PF, consistent with the proposed myofibroblastic spectrum of PF cell development.Calponin is a nonspecific muscular marker for differentiated smooth muscle cells, while CD10 indicates cells with fibroblastic traits. The calponin and CD10 results confirmed the variably myofibroblastic nature of PF in the muscular and fibroblastic axes, respectively. The ER showed all negative staining, but the PR were diffusely or focally reactive in 8 of 10 cases , 31, 59,β-catenin, ALK, cytokeratin AE1/AE3, and synaptophysin, thereby excluding epithelial, perineural, neuronal, and neuroendocrine cell lineage and ruling out some of the important differential diagnoses, such as fibromatosis and inflammatory myofibroblastic tumor.The rest of the markers mainly had negative results in PF. DOG-1 and CD117 were always negative in the reported cases, which allowed PF to be well-distinguished from GIST . CD34 wa C-KIT and PDGFRA gene mutations are important and characteristic in GIST [ glioma-associated oncogene homologue 1 (GLI1) and metastasis-associated lung adenocarcinoma transcript 1 (MALAT1) have been identified in a subset of PF cases [ GLI1 gene translocation reported in 6 cases (24%) and GLI1 polysomy reported in 2 cases (8%), out of overall 25 cases with GLI1 genetic analysis [ GLI1 oncogenesis may be sensitive to Hedgehog pathway inhibitors, targeting either Patched, Smoothened, GLI1, or other Hedgehog pathway components [ GLI1 mutation and Hedgehog signaling may bring about improved understanding of its pathogenesis and even novel therapeutic strategies.Genetic mutation has also been examined in some studies. The in GIST , but thePF cases , 59, witanalysis , 59, 78.analysis , 85 and analysis , which panalysis . Apart fanalysis . The molmponents . PF remaClinical findings are indecisive for the diagnosis of PF, as a solid, elastic tumor with or without ulceration is most likely to be encountered first during endoscopy, with typically nonspecific or hemorrhagic gastrointestinal signs or symptoms. Diagnosis of PF depends on pathological and immunochemical examinations, which are not achieved by clinical examination alone , and a m C-KIT or PDGFRA mutations of GIST are adopted widely in routine pathological practice [The most important differential diagnosis of PF is GIST, especially myxoid variants of GIST with spindle-cell histology , 89. Mospractice .Other pathological differential diagnoses, based on spindle cell morphology and myxoid stromal background, include sarcomatoid carcinoma, peripheral nerve sheath tumor, inflammatory fibroid polyp, and inflammatory myofibroblastic tumor. Primary or secondary sarcomatoid carcinoma with spindle tumor cells and myxoid stroma could be sometimes mistaken as gastrointestinal mesenchymal neoplasm. A clinical cancer history, a more malignant cytological feature, such as brisk mitotic or apoptotic activities, positive epithelial immunoreactivities, and markers for possible metastatic origins, such as TTF-1 for pulmonary origin, are significantly conducive to the correct diagnosis .Peripheral nerve sheath tumor includes benign schwannoma, neurofibroma or ganglioneuroma, and malignant peripheral nerve sheath tumor (MPNST); some variants also demonstrate a plexiform growth pattern . NeurogeInflammatory fibroid polyps consist of bland-looking spindle cells arranged in perivascular whorled pattern in a myxoedematous inflammatory stroma with mostly eosinophils. Positive CD34 immunoreactivity is the key characteristic of this rare tumor, which should be negative in PF except for vascular endothelial cells .Inflammatory myofibroblastic tumor is characterized by monotonous spindle cells arranged in fascicles or vague whorls in an inflammatory and edematous stroma full of neutrophils, eosinophils, or lymphoplasma cells. Although the myofibroblastic nature of an inflammatory myofibroblastic tumor is similar to that of PF by morphology and immunohistochemical studies, the lack of a plexiform growth pattern, a predominantly inflammatory cellular microenvironment, and positive ALK immunoreactivity help to distinguish inflammatory myofibroblastic tumor from PF .The mainstream treatment of PF is surgical removal, while medical treatment serves an assistant role for symptomatic management. Among the 121 reported cases, treatments were available in 99 cases and not provided in 22 cases. Partial gastrectomy (N=30) and distal gastrectomy (N=26) are the most frequently performed surgical treatments for PF, followed by local resection (N=12), subtotal gastrectomy (N=10), wedge resection (N=7), submucosal dissection (N=6), antrectomy (N=3), partial duodenectomy (N=2), nonsurgical follow-up with only endoscopic biopsy (N=2), total gastrectomy (N=1), and laparoscopic cholecystectomy (N=1). A laparoscopic operative technique was performed in 10 cases, endoscopic resection in 9 cases (including 1 thoracoscopic resection), and laparoscopic endoscopic cooperative surgery (LECS) in 4 cases. None of the surgically treated cases presented with severe postoperative complications, except for a 63-year-old male patient who had experienced recurrent GI bleeding for 17 years with a duodenal PF and had undergone pancreas-preserving duodenectomy of the first 2 portions, complicated by a low output pancreatic-skin fistula which had totally drained after 4 months. Secondary procedures included Billroth's operation I (N=3), Billroth's operation II (N=2), Roux-en-Y anastomosis (N=2), and omentectomy (N=1).Conservative management included endoscopic resection and nonsurgical intervention. Since PF is currently considered a benign disease (though not yet verified by convincing evidence), conservative management may be suitable for PF patients, and particularly for the elderly or selected patients with surgically contraindicated comorbidities ; at leasPF shows a benign biological behavior , 73, 74,Of all the 121 reported cases, follow-up was not reported in 37 cases, and the follow-up period was not reported in 4 cases. No malignant change, local recurrence, or PF-related mortality was reported. Most of the cases were uneventful after treatments, and among the cases that had follow-up periods, the uneventful or alive duration ranged from 0.75 to 396 months, with an average of 44.29±72.5 months and a median of 15 months. An 81-year-old female who underwent nonsurgical management also had her symptoms resolved 3 months after . Three cPF is a rare mesenchymal tumor with increasing clinical attention and occurs mostly but not exclusively in the GI tract. Typical clinical presentations are nonspecific GI signs or symptoms, or upper GI bleeding. Endoscopic biopsy is recommended for visualizing the microscopic features of PF with benign cytological traits, and immunohistochemical staining is required for diagnosis as well as for exclusion of GIST. Surgical local excision is the main treatment; however, more conservative management is suggested within an individually reasonable range because of its very good prognosis.


Background: Because reports on the management of recurrent granulosa cell tumor have been sparse, a consensus as to which patients should undergo surgical resection and which patients should be considered for chemotherapy has not been established. Methods: A total of 21 tumor recurrences in eight patients with granulosa cell tumor were reviewed. Results: Surgery was performed as the main treatment for 13 recurrences, while chemotherapy was chosen as the main treatment for eight recurrences. Complete tumor resection could be accomplished in 13 of 16 surgeries (81.3%), which include all the ten recurrences without involvement of liver or diaphragm and without ascites. The number of recurrent masses was significantly higher in the early recurrence group  than in the late recurrence . All cases with a solitary recurrent tumor at an extra-peritoneal site presented a significantly longer progression free survival. Conclusions: For patients with recurrent granulosa cell tumor, surgery may provide the best disease control. In cases with complete resection, the number of recurrent masses was the predictive factor for the next recurrence, and adjuvant chemotherapy might be considered in such cases. Granulosa cell tumor (GCT) is a rare ovarian malignancy which accounts for 2% of all ovarian malignant tumors . PatientThe management of primary GCT has been the topic of many reports. The recommended surgical procedure, indication for adjuvant chemotherapy, and choice of chemotherapeutic agent have been described in the national comprehensive cancer network (NCCN) guideline . HoweverRegarding the chemotherapy regimen for recurrent GCT, Bleomycin Etoposide Cisplatin (BEP) has been recommended based on the GOG trial . AccordiIn this study, we retrospectively reviewed 21 treatments for recurrent GCT in an attempt to find predictive factors for successful surgery among the indications for surgery. In addition, we also sought to identify prognostic factors after surgery to assist in determining the indication for post-operative adjuvant chemotherapy.A total of 21 tumor recurrences in eight patients with ovarian GCT treated at Kindai University Hospital between 1996 and 2010 were included in this study. As the primary treatment, unilateral salpingo-oophorectomy (USO) was performed at a minimum and the pathological diagnosis was confirmed by a gynecologic pathologist. Among eight patients, seven (87.5%) had repeated recurrences, with a total number of 21 recurrent events in these. Surgery was performed as the main treatment for 13 recurrences (surgery-group), while chemotherapy was chosen as the main treatment modality in the remaining eight recurrences (CT-group). In three of these recurrences, laparotomy was actually performed, but only tissue biopsy was sampled due to the presence of unresectable tumor; these cases were analyzed as part of the CT-group. In 16 recurrences (13 in the surgery-group plus three in the CT-group), the diagnosis of recurrent GCT was confirmed pathologically, while the diagnosis of recurrence was made only by computed tomography in the other five recurrences. In each recurrence, the patient characteristics, method of treatment, and progression-free survival (PFS) after treatment were analyzed.Participants were fully informed of procedures, and written informed consent was obtained from each. The ethics committee for human studies at Kindai University faculty of medicine approved the present study, which was conducted in accordance with the Declaration of Helsinki .2 and t tests for unpaired data. The Kaplan–Meier curve was determined by log-rank analysis of progression-free survival. All tests were two-tailed, and the results were deemed to be statistically significant at a level ≤0.05.Comparison of the categorical data was performed using the ΧThe clinical characteristics of the eight patients are shown in The median age of the patients was 44 years (range 32–68). For their primary lesion, seven patients were diagnosed as stage Ic, and one patient as stage IIb. As their primary surgery, five cases underwent USO, one case underwent abdominal total hysterectomy and bilateral salpingo-oophorectomy (ATH + BSO), and two cases underwent ATH + BSO with pelvic lymph-node dissection. None of them received adjuvant chemotherapy. The median recurrence-free survival was 56.5 months (range 14–168) and the median follow-up period was 135 months (range 101–349).The treatment course of the eight cases are shown in Recurrences were divided into three groups; (i) recurrence at abdominal cavity without liver/diaphragm involvement and without ascites, (ii) involvement (including surface) of liver/diaphragm with or without ascites, (iii) peritoneal dissemination with ascites. Surgeries were performed for all the ten recurrences without liver/diaphragm and without ascites, all of which achieved macrospically complete resection, followed by adjuvant chemotherapy in only one case (case 1). Of the eight recurrences involving the liver and/or diaphragm, chemotherapy was selected for four cases, none of whom achieved a complete response, and surgeries were performed for three cases, which included one complete (case 2) and two incomplete surgeries (case 3). One patient did not wish to receive anti-tumor treatment (case 7). Of the four recurrences with peritoneal disseminations associated with ascites, pre-operative chemotherapy was performed for two cases followed by complete surgeries (case 5 and case 7). One case received surgery followed by chemotherapy (case 8) and another case received only chemotherapy (case 4). The number of patients who survived without disease was four, three, two, and one at five, ten, twenty, and thirty years after the initial treatment, respectively. Regarding the intervals to the next recurrence, the median progression free interval from the treatment of the first recurrence to the second recurrence, second to third, and third to fourth was 21, 27, and 19 months, respectively, with no significant difference among them .Factors predictive of early recurrence were analyzed by comparing early and late recurrences .p = 0.013). In addition, the mean number of tumors was significantly greater  in the PFS < 2 years group than in the PFS > 2 years group  (p = 0.03). There was no significant difference in terms of mean size of tumor, mean number of previous recurrences, tumor rupture and positive peritoneal cytology, and use of adjuvant chemotherapy.Early recurrence was defined as a recurrence that occurred within 2 years after the previous recurrence (PFS < 2 years group) and late recurrence as that occurred more than 2 years after the previous recurrence (PFS > 2 years group). There was no significant difference between these two groups with regard to the site of recurrence, but in three recurrences in the PFS > 2 years group, the tumor recurrence involved the inferior rectus and oblique abdominal muscles , and comparatively long-term PFS were observed . In those three recurrences, the median PFS was significantly longer than the other 10 surgical treatment cases  in three treatment courses, gemcitabine in two, cyclophosphamide + cisplatin in one, and cyclophosphamide in one. The mean number of chemotherapy courses was 9.9 ± 7.9 (range 2–26). The overall response according to the revised response evaluation criteria in solid tumors (RECIST) guideline ver. 1.1 was 62.5% (5/8), and a complete response was observed in 25% (2/8). The median PFS was 38 months and the median overall survival was 48 months. Adverse events according to CTC-AE ver. 4.0 are shown in A good recovery was noted for all the adverse events and there was no treatment-related death.Management for primary GCT in terms of method of surgery, efficacy of adjuvant chemotherapy, and adjuvant radiotherapy has largely been standardized by the previous studies ,14,15,16Whether surgical resection of recurrent GCT is possible or effective was first evaluated. In this study, the number of recurrent tumors per case was greater than three in all treated cases. However, complete resection could be accomplished in 13 of 16 surgically treated cases (81.3%), including a case with 16 recurrent masses. The size of tumor, mean number of recurrent masses, mean number of recurrences, and mean interval from the previous recurrence did not influence the choice of surgery. Likewise, in the literature, more than half of recurrent GCT has been reported to occur at multiple sites. The optimal debulking rate has been assumed to be as much as 80–85%, and optimal debulking is considered to improve prognosis ,21,22. IIn the case of epithelial ovarian cancers, the intervals to second and third time of recurrence tend to be shorter than the previous ones and the treatment becomes more difficult with repeated recurrence. By contrast, in our study, there was no significant difference in the intervals as recurrences occurred. Fotopoulou et al. reported in 27 recurrent GCT cases that the median PFS from the first to the second recurrence was 20 months, while that from the second to the third recurrence was 18 months . In the Another important issue in the management of recurrent GCT is the use of adjuvant therapy after surgery, about which very few reports have appeared. The MITO-9 trial did not recommend post-operative adjuvant chemotherapy for recurrent GCT because 11 of 35 31.4%) cases recurred despite post-operative adjuvant therapy being performed in 81.8% of all cases. However, in most of the previous studies, as much as 70–80% of GCT recurrences recurred again and most of these patients died of disease % cases r. By compTo determine which case actually would be the indication of adjuvant therapy, we investigated predictive factors for recurrence after surgery in recurrent GCT. We compared the patient characteristics between cases with late recurrence after previous treatment (PFS > 2 years) and those with early recurrence (PFS < 2 years). No significant difference could be found in the site of recurrence, the size of tumor, the number of recurrences, tumor rupture, and positive peritoneal cytology. In contrast, the number of recurrent masses was significantly higher in the early recurrence group, suggesting the residual presence of micro metastatic foci in the peritoneal cavity. Therefore, post-operative adjuvant therapy may be considered particularly in cases with multiple intra-peritoneal recurrences. In contrast, all cases with a solitary recurrent tumor at an extra-peritoneal site presented a significantly longer PFS. These recurrences may represent implantation in a surgical wound, which has been occasionally noted in GCT . In thesIn this study, various chemotherapy regimens were used, including BEP, TC, CP, cyclophosphamide, and gemcitabine. The standard chemotherapy for recurrent GCT is BEP therapy based on the phase II GOG trial  and PVB In summary, for patients with recurrent GCT, surgery may provide the best disease control compared with other treatments, and optimal surgery could be accomplished in most of the cases. In cases with complete resection, the number of recurrent masses was the predictive factor for the next recurrence, and adjuvant chemotherapy might be considered in such cases. With regard to chemotherapy regimens, the less toxic regimens including gemcitabine may be considered because long-term management is necessary in many cases of recurrent GCT due to repeated recurrence. Because a randomized trial may not be possible for this rare disease, the continued accumulation of treatment experiences is needed.


Nevus of Ota or “oculodermal melanocytosis” is a rare congenital hamartoma of dermal melanocytes causing a blue-gray hyperpigmentation of the eye and surrounding structures. The condition, originally described by Ota and Tanino in 1939, mainly affects the ophthalmic and maxillary divisions of the trigeminal nerve. We describe the first reported case of unilateral oculodermal melanocytosis in a Caucasian woman with oral buccal mucosal involvement. Oral involvement of nevus of Ota is very rare.A 48-year-old Caucasian woman was referred by the dermatology division to the oral medicine department at the University of Liverpool School of Dentistry with new-onset oral pigmentation to the left buccal mucosa. The patient had a previous diagnosis of oculodermal nevus.An incisional biopsy of the left buccal mucosa was completed. The report stated that histological and immunohistochemical features were in keeping with a blue nevus, but within the context of the preexisting occulodermal pigmentation, a diagnosis of oculodermal melanocytosis, also known as “nevus of Ota,” was made. The patient will be kept under review in the oral medicine department because the progression of the lesion on the left buccal mucosa requires active monitoring owing to the potential for malignant change. The patient also requires regular review in the dermatology and ophthalmology divisions. Nevus of Ota or “oculodermal melanocytosis” is a rare congenital hamartoma of dermal melanocytes causing a blue-gray hyperpigmentation of the eye and surrounding structures . OriginaA 48-year-old Caucasian woman was referred to the oral medicine department at the University of Liverpool School of Dentistry with new-onset oral pigmentation to the left buccal mucosa. Her past medical history revealed a diagnosis of “oculodermal nevus.” She recalled having pigmentation in her left eye from birth and pigmentation of skin of the left face since the age of 13 years, for which she received laser treatment for cosmetic purposes. The patient also reported annual monitoring of a benign intracranial tumor along with close monitoring by ophthalmology and dermatology divisions. She did not take any regular medications. She did not smoke or consume alcohol. She is single and lives on her own with no dependents. She lives close to her mother, who attended the appointments with her.On examination, a subtle but diffusely speckled bluish pigmentation was observed to the left midface involving the infraorbital and zygomatic regions. A post–laser therapy yellow hue was noted on the left periorbital skin. Pigmentation of the sclera and conjunctiva was also observed. Intraorally, an inhomogeneous, blue-gray, diffuse hyperpigmentation affecting the entire left buccal mucosa was noted. Mild pigmentation of the left hard palate was also noted Figs. , 4 and 5An incisional biopsy of the left buccal mucosa was completed. The report stated that histological and immunohistochemical features were in keeping with a blue nevus, but within the context of the preexisting occulodermal pigmentation, a diagnosis of oculodermal melanocytosis, also known as “nevus of Ota,” was made. No other investigations were required. The patient will be kept under 6-monthly review with the oral medicine department because the progression of the lesion on the left buccal mucosa requires active monitoring owing to the potential for malignant change.Nevus of Ota represents a unilateral dermal melanosis in the distribution of the trigeminal nerve, where it is usually confined to the ophthalmic and maxillary divisions. The forehead, temple, periorbital area, cheek, and nose are most commonly involved. Melanin pigmentation involves the eye in about 50% of cases . Rarely A literature search of PubMed and Embase revealed only two cases (in India) of nevus of Ota affecting the buccal mucosa , 6. IntrThe prevalence of this condition is greatest in the Asian continent, affecting up to 1% of the Japanese population . One stuUse of lasers for treatment of nevus of Ota has become helpful in the management of dermal nevi. Currently, Q-switched lasers are the most studied, and they have demonstrated positive results for nevus of Ota . Lasers GNAQ and BAP1 genes [Our patient had received laser therapy 20 years earlier and had a good outcome. She has also been referred to the cosmetic camouflage clinic. The patient has seen a makeup artist and uses cosmetics to cover the pigmentation. Long-term follow-up, especially in the ophthalmology division (6-monthly)  and neurP1 genes . This wi


ICR). The inhibition of ICR progression and environmental improvement in and around the affected tooth through combined endodontic and periodontal treatments led to a favorable clinical outcome.Dental radiography and cone‐beam computed tomography revealed the left mandibular first molar in a 68‐year‐old female patient with Heithersay Class 3 invasive cervical resorption ( This externally resorptive process is characterized by a cervical location and leads to progressive and destructive loss of tooth structure. The etiology of ICR is unclear, however, mechanical, inflammatory, autoimmune, or infectious stimulus are considered predisposing factors of ICR.The cases of anterior teeth outnumber those of posterior teeth in the treatment of ICR.2A 68‐year‐old female patient visited Hiroshima University Hospital with a chief complaint of gingival discomfort around the left mandibular first molar tooth 36). She had no history of trauma, orthodontic treatment, or bleaching, however, the affected tooth and the second premolar were abutments of a three‐unit metal cantilever bridge. The pontic, which was connected to the two crowns, extended into the missing second molar space. This bridge had been fixed approximately 15 years before the first visit. She had a malocclusion, open bite, and crossbite   and root canal treatment were performed with the patient's informed consent.The full thickness flap was raised to allow visualization of the entry point of the granulomatous Figure A, which The patient was recalled 1 week after the operation. The tooth had been asymptomatic. The tooth was isolated with a rubber dam. After removal of the temporary seal Figure G, the roOne month after root canal filling, thin attached gingiva and plaque accumulation on the cervical contour were observed in tooth 36 with the temporary crown Figure A. To incThe 3‐year follow‐up demonstrated that the tooth with the full metal crown exhibited no clinical symptoms  and adequate width of attached gingival was maintained Figure A compare3In some Heithersay Class 2 and Class 3 cases, the lesion is very close to the pulp and dentin between the lesion and the pulp is very thin. Therefore, endodontic treatment may be necessary since a risk of perforation into the pulp cavity exists upon removing the granulomatous tissues.There are several successful case reports using nonsurgical approaches, such as topical application of 90% aqueous solution of trichloroacetic acid (TCA) to the granulomatous tissues, orthodontic extrusion, and low‐power neodymium‐doped yttrium‐aluminum‐garnet (Nd:YAG) laser irradiation.Amalgam, composite resin, glass ionomer cement, resin‐modified glass ionomer cement, and MTA are available to fill the defect during surgical treatments, such as apically positioned flap and repositioning flap.The fundamental objective of endodontic treatment was to eliminate bacteria in the root canal system because they play an important role in the onset and development of periapical lesions.In this case, since attached gingiva had been lost and, as a result, plaque had readily accumulated at the buccal cervical contour of tooth 36 after the first surgical treatment, FGG was performed at the affected tooth to gain attached gingiva.4The diagnosis of ICR using CBCT, perforation, and defect repair by filling with MTA at the diseased site, endodontic treatment with bacterial examination and a healthier oral environment by FGG contributed to a favorable clinical outcome in this case. Thus, this case report has revealed that combined endodontic and periodontal treatments have value in treating ICR of a molar.None declared for all authors.TN, KT, YA, TI, RY, HK and HS: drafted the manuscript and contributed to treatment of the patient. All authors have read and approved the final manuscript.


The odontogenic keratocyst (OKC) may occur at any age. However, it mostly occurs during the second and third decades of life. Compared to other odontogenic cysts, this type occurs with a frequency of 5-15%. It is more common in the mandible region and in the male sex. Histologically, odontogenic keratocysts are characterized by the presence of an external connective tissue capsule, with keratinizing lining of the epithelium consisting of 5-8 cell layers with marked palisadisation of polarized basal cells and a corrugated parakeratin layer. The objective of this study is to present cases of odontogenic keratocysts, with reference to the latest classification and dilemmas in therapeutic doctrine. This project was realized in the form of descriptive studies, specifically in a series of cases. A collection of four individual cases was found at the Department of Oral Surgery. Due to the proper approach towards diagnosis, adequate and detailed histopathological analysis, and suitable therapeutic procedures, all cases of odontogenic keratocysts were successfully treated without complications. Enucleation of OKC, with a regular follow-up, proved to be the effective therapeutic choice for the patients described in this paper. Only in the case of recurrence would we consider other therapeutic options, primarily enucleation in combination with Carnoy's solution. The odontogenic cysts are a heterogeneous group of lesions which are, according to the new WHO nomenclature, classified into odontogenic inflammatory cysts  and odontogenic and nonodontogenic developmental cysts . In contAccording to the literature, OKC is more common in the mandible, in the lateral region, with the highest incidence in patients being between ten and thirty years of age . It showFrom 1950 to 2017, the classification of odontogenic keratocysts (OKCs) underwent substantial changes. Chronologically, the name “keratocyst” was first introduced in 1950. The same name was officially used in the WHO classification from 1971 to 1992. According to these classifications, there were two subtypes of odontogenic keratocyst (OKC): the parakeratinized and orthokeratinized types. However, in 2005, these two types were classified as independent entities, mostly because of the differences in their tendency to recur. The parakeratinized type was classified as a “keratocystic odontogenic tumor” (KCOT) under the branch of epithelial tumors of odontogenic origin. The orthokeratinized type was classified as an orthokeratinized odontogenic cyst (OOC) as part of the entity of odontogenic developmental cysts. According to the newest WHO classification from 2017, keratocystic odontogenic tumors (KCOT) were again reclassified as odontogenic keratocysts (OKCs) but still diagnostically distinctive from orthokeratinized odontogenic cysts (OOC) .PTCH1 gene mutations were found in other developmental cysts [One of the reasons for the WHO's reintroduction of the term “odontogenic keratocyst” (OKC) was because al cysts . Anotheral cysts , 5, 12. al cysts –16.The origin of OKC is not quite clear. It is presumed to be either primordial, including dental lamina remnants, basal cells of the overlying epithelium, or dentigerous, which implies reduced enamel epithelium of the dental follicle .In this paper, the authors present 4 interesting cases from their own practice. The cases described are particularly interesting for their clinical characteristics  and differential diagnosis. The paper also contains a mini literary review, in which the authors refer to the latest classification of odontogenic keratocysts and their therapeutic possibilities.2.1.A 10-year-old patient arrived at our clinic due to a tumefaction in the frontal region of the maxilla, especially above the deciduous canine on the left. Clinical examination revealed deformity of the maxilla in that region, in the form of a round swelling. The bone in the described area had been reduced to egg-shell thickness, and a crackling sensation was felt on pressure (Dypitren's phenomenon). There was no pain. Inspection also revealed the presence of an ectopic lateral permanent incisor on the left .On the orthopantomogram (OPG) image, ellipsoidal, longitudinally positioned radiolucency was observed, which diverged at the roots of the adjacent teeth, i.e., the lateral incisor and the deciduous canine. In the apical part of this radiolucency, the crown of the retained permanent canine was prominent . AccordiUnder local anesthesia, a trapezoidal flap was raised. Subsequently, it was noticed that the cyst had completely expelled the bone fragment from the vestibular side as it grew. The thinned, vestibular bony wall was carefully cut off and removed. Thus, the boundaries of the cyst, containing a protective white outer shell, were displayed . The cysHistopathological analysis of the removed cyst revealed characteristics of odontogenic keratocysts rather than dentigerous cysts as originally believed on clinical examination and radiographic diagnostics. The histopathological results had also showed the characteristic corrugated lining of parakeratotic epithelial cells on the luminal surface of the cyst wall, with a palisaded hyperchromatic basal cell layer supporting and strengthening the fibrous wall of the cyst .The follow-up period is 9 years. The patient has no symptoms and no clinical or radiological signs of recurrence .2.2.The patient came to the Oral Surgery Department for the surgical removal of an upper impacted third molar on the left. The patient stated that she had had swelling in the region several times and she had been repeatedly under antibiotic therapy.The crown of the impacted tooth 28 was located above the root of tooth 27 with obvious radio-opacity of the left maxillary sinus when compared to the right side .After application of local anesthesia, a triangular flap was raised and the bone exposed. After corticotomy of the vestibular side above the apex of tooth 27, the crown of tooth 28 was shown. During the extraction of tooth 28, there was a leak of thick, dirty yellow content that filled a large cystic lesion. The cyst was carefully enucleated . Its dimThe results of histopathological analysis indicated that the site was covered with a multilayered, parakeratotic, squamous stratified, lining epithelium with a preserved basal cell layer. Infrequent inflammatory infiltrates, rich in lymphocytes and plasma cells, could be detected in the subepithelial parts . HistopaThe follow-up period is 6.5 years with no radiological signs of recurrence (arrows indicate the area of interest) .2.3.The patient came to the Department of Oral Surgery at the Faculty of Dental Medicine, University of Sarajevo, for fenestration of vestibular mucosa in the tooth 34 region and edema and mandible pain in region 34-43. Anamnesis revealed the outpatient removal of a semi-impacted tooth 35 years ago . ClinicaCBCT analyses Figures  showed wAfter application of local anesthesia (V anesthesia and left mandibular block), a trapezoidal flap in the region of 34-43 was raised. It was noted that the lesion had destroyed the vestibular bone in regions 31 and 41 Figures . The exiThe microscopic finding showed that on the surface of the sample there was a uniform multilayer epithelium without the presence of rete ridges. The basal cell layer was characterized by remarkable hyperchromatic nuclei. The luminal surface was corrugated and parakeratotic. In the cystic wall, there were epithelial cell invaginations that had produced satellite cysts and chronic inflammatory infiltrates. Histopathological analysis confirmed the diagnosis of odontogenic keratocyst .Two years after the second operation, the patient did not follow through with regular checkups so data about possible recurrence are not available.2.4.The patient visited the Oral Surgery Department due to a swelling in the frontal vestibular region of the upper jaw above the central incisors . A cliniA large radiolucency that diverged at roots 11 and 21 was visible on the orthopantomogram (OPG) X-ray .Following the application of local anesthesia, a trapezoidal flap was raised. After that, it was observed that the vestibular part of the bone had been destroyed along the width of the lesion (2.5 × 2.0 cm). A noticeable cavity in the bone was filled with dense yellow content . After tHistopathological analysis showed the characteristic histological image of a keratocystic odontogenic tumor with a multilayer epithelium of 5-8 cell thickness and a corrugated and parakeratinized surface, with the absence of inflammatory cell infiltrates in the underlying supportive connective tissue .The follow-up period is 1 year. The patient is symptomless with adequate signs of bone regeneration evident on X-ray analysis Figures .The classification of odontogenic keratocysts (OKCs) is still a topic commonly discussed by many dental researchers. According to the most recent WHO classification, the preferred term remains odontogenic keratocysts due to the lack of evidence of its neoplastic nature . This obIn all the histopathological findings in our described cases, the existence of a corrugated parakeratotic layer of epithelial cells was emphasized, which is a basic feature of OKC. For this reason, we consider that histopathological analysis is imperative in setting the diagnosis of these cysts. In other words, the obligation of each surgeon is to perform a biopsy and send the sample to a pathologist for analysis. Given that the finding of parakeratosis is linked to a tendency to relapse, all our patients were warned of this possibility and advised that regular controls are crucial. The follow-up period of the described cases ranged from 1 year to 9 years. In the opinion of a large number of authorities, it usually occurs 2 years postoperatively, so OKC detected histopathologically should be monitored for at least 5 years postsurgery [Although OKCs are strictly diagnosed only by histological examination, there are some radiological and other features that can be of help in approaching the diagnosis, with an indication that these features are not pathognomonic for odontogenic keratocysts . For exaThe radiological appearance of OKC is unilocular and multilocular. The same appearance can be seen in other jaw lesions of odontogenic and nonodontogenic origin. This is important in differential diagnosis where OKC may be misdiagnosed as other odontogenic and nonodontogenic cysts and ameloblastoma . UnilocuOKCs, especially large ones, show a specific pattern of growth. Large mandibular OKCs grow in a mesiodistal direction along the length of the bone, with minimal buccolingual expansion. This type was also recognized in Case #3. An OKC located in the maxilla reveals expansion of the cortical bone, which can be seen as a bone deformity . This chOne of the consequences of the described growth pattern of OKC in the upper and lower jaws is that these cysts are usually asymptomatic for a long period of time. Patients usually seek help from doctors if deformities in the bones are noticed (Cases #1 and #4) or if spillage of cystic pus (Case #2) or a fistula is present (Case #3).Of the other clinical features, OKC cystic fluid is also important. This content can be of different viscosities and colors, from straw yellow and thin to cheesy and thick. For Case #2 and Case #3, this content was dense in consistency and dirty yellow in color, and in Case #4 light yellow and murky. In Case #1, the content of the cyst was not a dominant symptom in the clinical findings. In addition to the question of classification, it is even more interesting to consider the therapeutic possibilities for OKC. There are two methods in the treatment of these lesions, one conservative and the other aggressive , 28–37. The reported recurrence rates for OKC vary from 5% to almost 70%, depending on the therapeutic procedure . There aIt is certain that an aggressive and radical approach to therapy will lead to the reduced possibility of recurrence, but the question arises: what if we have a young patient who is still in the phase of eruption of permanent teeth? By aggressive therapy, the integrity of the jaw bone would be violated and the procedure would practically mutilate the patient without any certainty as to whether there would be recurrence in general . In the Dong et al. conclude that children with such cysts and with permanent teeth still to erupt should be treated conservatively rather than aggressively, since aggressive treatment may have a negative effect on tooth development, eruption processes, and jaw development . On the α and cytokeratin-10 that are thought to be related to cyst expansion. In addition, after decompression and marsupialization, the epithelial lining converts in a less aggressive form. However, both of these methods imply the longer duration of therapy, a greater number of interventions, and in particular the patient's cooperation. It is also possible that the biopsy sample that is taken during decompression and marsupialization is not representative [Pitak-Arnnop et al. consider that decompression and marsupialization are useful in some cases . Both meentative .Pitak-Arnnop et al. devised an algorithm in 2009 for the treatment of OKC and other cyst-like lesions. Thus, regardless of the histological types and localization of lesions, they favor enucleation in combination with adequate postoperative control , 13.It is ideal to remove the cyst in one piece. If the cyst is located periapically or if access is difficult and if it is a recurrent cyst, peripheral ostectomy and/or curettage is recommended. After both approaches, histopathology is obligatory. If an odontogenic keratocyst is confirmed, this does not mean that the surgery should be repeated immediately because 25-50% of cases recur after a simple enucleation. Thus, a “Wait and see!” protocol should be applied [Considering that in the differential diagnosis of OKC there are follicular-dentigerous cysts and ameloblastomas, in the therapeutic approach enucleation should not be avoided in order to remove the lesion in one piece. This would be our attitude, based on the doctrinal approach of many authors .Many have tried to answer the question regarding which factors influence OKC recurrence.It is still not known exactly where the epithelial islands and microcysts are located. They could be localized in a cyst capsule, in the overlying soft tissue, and/or in the bony bed of the cyst. Carnoy's solution and liquid nitrogen are recommended for epithelial island and microcyst elimination , 44. It As recent research has shown that most of the epithelial islands and/or microcysts are located in the mucosa that is directly in contact with the cyst, it is not necessary to remove and freeze the adjacent bone tissue. Moreover, peripheral ostectomy may disperse microcysts that are immersed in the bone and thus contribute to a higher risk of recurrence , 43.Over the past few years, a great deal of attention has been dedicated to new procedures in the treatment of OKC in order to make it as successful and simple as possible, thereby improving the final outcome of the therapy. One of the therapeutic agents considered in this procedure is an antimetabolic drug, 5-fluorouracil, which is believed to induce apoptosis of the cells in a hepatocellular carcinoma. In addition, 5-fluorouracil has other domains of use, such as its topical application in the treatment of basocellular carcinoma and keratocystic odontogenic tumors. For 5-fluorouracil, it is considered to be more acceptable in the treatment of a keratocystic odontogenic tumor than Carnoy's solution, due to its availability, technical simplicity, shorter operating time, and equally successful, reduced morbidity of other tissues compared to Carnoy's solution. In order to use it, it is necessary to soak the gauze in a 5% solution of this drug, which is then applied to the residual bone cavity so that it may easily be found 24 hours after surgery. In contrast, Carnoy's solution requires a much longer period of time when it comes to its use, given the fact that it is necessary to comply with the many precautions that this product requires. In addition, with the use of 5-fluorouracil, no side effects of the topical application were noticed in the study carried out by Ledderhof and associates. However, systemic administration of 5-fluorouracil may lead to certain side effects, such as mucositis, agranulocytosis, neuropathy, nausea, vomiting, hypotension, exhaustion, and even death. Death is usually due to a lack of enzyme dihydropyrimidine dehydrogenase (DPD) that breaks down this drug resulting in systemic toxicity. Most commonly, this enzyme is missing in African woman with a frequency of 12%, which should be considered in the systemic administration of this drug .In the last few years, studies have also demonstrated the success of cyclopamine, a plant alkaloid, which inhibits cell response to the SHH (sonic hedgehog) signal. Cyclopamine blocks the activation of the SHH signaling pathway, thereby proving effective in treating OKC. This therapeutic possibility has not yet been fully developed, due to the lack of data on its success, as well as the fact that it requires research and action on the molecular level .In all our described cases, a conservative therapeutic approach was adopted in the form of cyst enucleation without the use of Carnoy's solution or any other additional substance. The reason for this approach is based on the fact that Case #1 and Case #2 were primarily considered to be dentigerous cysts, which was why enucleation was planned as the only therapeutic option. Since histopathology diagnosed OKC, the patients are regularly monitored. In a case of suspected relapse, reenucleation with the use of Carnoy's solution would be shortlisted. In Case #4, the patient mentioned a traumatic blow in the region of the upper frontal teeth where the cyst was localized. This time a radicular cyst of traumatic etiology was suspected where enucleation was also the first choice. The patient is still being monitored regularly. As in previous cases, reoperation in the form of enucleation using Carnoy's solution will only be considered in the case of recurrence. Of all these cases, Case #3 was first suspected of having an OKC, primarily due to the radiological characteristics, localization of the lesion, and the age of the patient . The authors want to emphasize that at the time of preparation for surgery, they were not in possession of Although there are many therapeutic approaches for this type of cyst found in literature, there is still a lack of a universal therapeutic approach , 29, 47.The advantages of a conservative approach include fewer morbidities than radical resection . The disIn terms of differential diagnosis, in the case of younger patients (children and adolescents), when considering a dentigerous cyst, one should refer to odontogenic keratocysts. Enucleation of OKC with regular follow-up proved to be an adequate therapeutic choice for the patients described in this paper. Only in the case of recurrence would other therapeutic options be considered, such as enucleation in combination with Carnoy's solution.


CMUSE is a rare disease whose diagnosis remains difficult because the lesion is confined to the small bowel.Here, we present a case of 43-year-old female patient suffered chronic abdominal pain for 20 years, and finally diagnosed with CMUSE. Capsule endoscopy was performed when general endoscopic investigation failed to find the lesion, but the capsule was stranded in the small intestine. Moreover, capsule retention results in acute intestinal obstruction. Thus, surgery was performed and CMUSE was confirmed. The patient was recovered after partial small intestine resection.Capsule retention occurred in nearly 60% of patients with CMUSE. Capsule endoscopy should be avoided when the patient is suspected of CMUSE, especially with severe anemia and radiologic finding in the ileum. Cryptogenic multifocal ulcerous stenosing enteritis (CMUSE) is a rare disease with unknown etiology and pathophysiology. This disease is an independent entity characterized by chronic and intermittent bouts of moderate ileus resulting from multiple short stenoses of the small bowel with shallow ulcers –3. It haA 43-year-old woman with remittent abdominal pain, dizziness, and fatigue for 20 years was admitted to our hospital in May 2018. She was also suffered from iron deficiency anemia since her adolescence. There were positive results of stool occult blood test but no evidence of bleeding in gastroscopy and colonoscopy during the progression of the disease. On examination, she had lower limbers edema and mild abdominal tenderness around the umbilicus. Vital signs were within normal range.Her past medical history included appendectomy, oophorocystectomy, and caesarean section. She denied NSAIDs taking. Laboratory examination confirmed iron deficiency anemia  and hypoalbuminemia level , C-reaction protein  was elevated. In addition, the fecal occult blood test was positive. Other test results, such as renal and liver functions, autoimmune antibodies, T-spot were within normal range. Meanwhile, colonoscopy and gastroscopy showed normal results. However, abdominal CT scan revealed segmentally thickened small intestinal walls but no mention of stenosis Fig. . Double Unfortunately, exploratory laparotomy was performed because of acute intestinal obstruction which may be induced by retained capsule after 2 weeks. During the operation, multiple segmental stenoses of the small intestinal were observed and the endoscopy-capsule was found in the ileum. A section of the small intestine, about 60 cm in length, was removed. There were approximately 26 circumferential superficial ulcers with strictures and the minimal distance between two strictures was about 1.5–3.5 cm Fig. . PostopeSince Debray et al.  reportedCMUSE is a chronic and recurrent disease. The most common symptoms include abdominal pain, anemia, and gastrointestinal bleeding , 5. DiagIn order to directly observe the lesions in the small intestine, both CE and DBE are viable –22. GeneCapsule retention is a potential disadvantage of intestinal obstruction which may require endoscopic or surgical retrieval of the capsule , 34. MorAs for treatment, the most effective is immunotherapy , 3 and s


Necrosis of the falciform and round ligaments is extremely rare, thus making the diagnosis challenging. It is often misdiagnosed as gallbladder pathology due to the presenting symptoms. Due to the rarity of this pathology, there is limited literature available.A 53-year-old white man presented to our hospital with signs and symptoms of gallbladder pain but turned out to have the rare entity of necrosis of the falciform and round ligaments. An extensive review of the world literature was performed using PubMed. Manual cross-referencing of reference lists was performed to obtain all available articles. The personal operative log of the senior author was also searched to reveal one additional case. Statistical analysis was descriptive only, given the small number of reported cases. Thirty-nine articles were found, among which forty-three case were identified, and one additional case was extracted from the operative log of the senior author. Unlike previous reports, we found that isolated inflammation and necrosis of the ligaments occurs at nearly the same frequency in both men and women, not predominantly in women as previously reported in smaller series. The mean age at presentation was 59.5 years old, and cases were typically initially diagnosed as gallbladder pathology, most commonly acute cholecystitis. Computed tomography more frequently than ultrasound revealed the falciform and round-ligament pathology.Isolated falciform and round-ligament inflammation and necrosis is a rare condition that is difficult to diagnose because it can present mimicking a wide variety of intra-abdominal pathologies, particularly gallbladder pathologies. It is often best treated by laparoscopic resection. Unlike prior reports, our review of the literature, which is the largest that we know of to date, shows that males and females are equally affected. Greater awareness of this entity will aid in future diagnosis. It is a derivative of the embryonic ventral mesentery.The falciform ligament is a triangular, or falciform, fold of peritoneum stretching between the umbilical fissure of the liver and the anterior abdominal wall. The free edge of the falciform ligament is the round ligament  showed fat stranding extending from the umbilical fissure, along the course of the falciform and round ligaments, anteriorly to the abdominal wall and inferiorly toward the umbilicus Fig. . UltrasoUpon admission, the patient was treated empirically with piperacillin and tazobactam 3.375 g intravenously every 6 hours, and in addition, he was given intravenous hydromorphone and ondansetron as needed for pain and nausea, respectively.Given his progressively worsening symptoms, he was taken for laparoscopic exploration. At operation, the falciform and round ligaments were severely inflamed and thickened, with adherent omentum Fig. . The falPathologic examination revealed hemorrhagic fat necrosis and fibrin thrombi of the omentum, with hemorrhage and inflammatory changes of the falciform and round ligaments. The gallbladder pathology revealed only minor, microscopic evidence of chronic inflammation and cholesterolosis.The patient’s postoperative outcome was good, and he was discharged on the second postoperative day. He failed to return for a postoperative follow-up visit, but 1 year later, he presented to the emergency room of our hospital with unrelated complaints but no abdominal pain.3. Imaging revealed fat stranding around the falciform ligament but no sign of acute cholecystitis. After an initial attempt at nonoperative management, the patient was brought to the operating room for diagnostic laparoscopy, given his persistent pain that was constant and located increasingly in the epigastrium. Laparoscopic exploration revealed isolated ligament necrosis of the falciform and round ligaments. This case is important to present for several reasons. First, it draws attention to the tendency of necrosis of these ligaments to mimic gallbladder disease. Second, it is unique in that the patient also was found to have a concomitant severely reduced gallbladder ejection fraction on a hepatobiliary scan. It is also noteworthy because, despite the availability of US and CT, this diagnosis of necrosis of the falciform and round ligaments was not recognized. Upon retrospective review of the patient’s CT scan, however, falciform ligament inflammation was appreciated, highlighting an important learning opportunity. One reason it was not fully appreciated initially was a lack of awareness of this entity, due both to its rarity and to the paucity of publications on spontaneous necrosis of the falciform and round ligaments. Finally, another important point highlighted by this case is that the treatment can be simple and effective via laparoscopic resection of falciform ligament.We present a case of a 53-year-old man with a 4-day history of epigastric pain with associated abdominal tenderness. His laboratory values were significant for leukocytosis of 15,000/mmIn addition to reporting this case, we performed a literature search in PubMed  using keywords “falciform ligament necrosis,” “inflammation,” “round ligament,” and “ligamentum teres.” This review revealed further important findings, including that this entity occurs at nearly at the same frequency in both men and women, not predominantly in women as previously reported in smaller series.Titles, abstracts, and full-text publications were obtained and screened for presentation of case reports of round or falciform ligamentitis. No language restrictions were applied. Full-text versions of all foreign articles were obtained. The following data were extracted from the selected articles: demographics, medical and surgical history, surgical technique used, and pathology findings. Screening of non-English papers was performed using standard web-based programs, with translation and confirmation performed by colleagues fluent in those languages. Data were summarized descriptively.This literature search yielded 43 case reports in 39 publications, with an additional 2 cases derived from a manual search of references, but these were excluded because they were pediatric cases, and 1 additional case was found in the senior author’s operative log, for a total of 44 cases after excluding pediatric cases. The included reports were published between 1976 and 2014. The articles retained for this review were published in English (25 articles), French (6 articles), Danish (1 article), German (2 article), Japanese (1 article), and Russian (4 articles).According to this literature review Table , round aAmong the reviewed cases, four patients had a past medical history of hypertension (including our patient), five patients had diabetes mellitus , two patients had previous choledocholithiasis, and one had cholangitis due to periampullary carcinoma. There was no mention of coronary artery disease, except in one patient. There was no clear correlation between any medical condition or prior surgery and incidence of falciform ligament inflammation.The most common presenting symptom was right upper quadrant and epigastric pain, as well as fever, and the most common physical examination finding was tenderness in the epigastrium and the right upper quadrant, with peritoneal signs being reported in three cases. Laboratory values were reported in only four articles, and inconsistently, precluding any meaningful analysis of laboratory values.Regarding diagnosis, most patients received their diagnosis intraoperatively. The vast majority underwent US of the right upper quadrant and CT. The most common presumed etiology for this presentation was gallbladder disease, with 12 such cases of gallbladder mimicry: 7 were cases of suspected acute cholecystitis, 4 cases were cholelithiasis, and 1 was a probable acalculous cholecystitis. Other common preoperative differential diagnoses were acute pancreatitis and diffuse peritonitis secondary to perforated duodenal ulcers. A rarer etiology was small-bowel obstruction secondary to epigastric herniation. Only one of all the reviewed cases had round-ligament necrosis as a primary diagnosis [US imaging typically revealed hypoechoic infiltration of the round ligament or infarction of the fatty appendage of the ligamentum teres suggestive of acute appendagitis of the ligament. Moreover, few cases of hepatic and biliary lesions and masses captured by US were reported , 3. CT wRegarding treatment, in a majority of the cases, surgeons opted for an open surgical approach over a laparoscopic approach; such an option was presumably available for a majority of the cases, because only eight articles were published before 1990. Among the reviewed cases, 22 patients underwent laparotomy; of these, 9 had laparoscopic procedures, and 7 were treated with medical management only, giving a ratio of 2.5:1. However, this ratio decreased for more recent cases (over the past decade); among cases published after 2009, there were 7 cases of open procedures and 5 laparoscopic surgeries (including our patient’s case), giving a ratio of 1.4:1. Cholecystectomy was performed in addition to the partial or total resection of the falciform ligament in eight (18%) of the analyzed cases.The most common intraoperative finding was partial or total necrosis or gangrene of the round ligament with abscess, which was reported in 24 55%) cases, and in a majority of cases, this was confirmed by postoperative pathology. Involvement of the gallbladder in the inflammatory process was noted in four cases 10%). There was only one mention in the analyzed literature of involvement of the liver 0%. There–10. Tumo5% cases,Inflammation of the falciform ligament can be explained by infection of surrounding structures such as the gallbladder, liver, peritoneum, and thoracic or abdominal wall. Isolated inflammation of the falciform ligament has also been reported. In addition, torsion of preexisting cysts in the falciform ligament can lead to infarction and gangrene , 13. EngMost commonly, as in our patient’s case, the falciform ligament and the round ligament are simultaneously inflamed. This may be attributed to the arterial and lymphatic supply to the ligaments. The right hepatic artery gives off a branch, which enters the round ligaments and gives off two to three branches that anastomose with tributaries from the superficial inferior epigastric artery. The venous flow is through the paraumbilical veins. The superior veins drain the medial diaphragm and traverse the upper and lower veins to communicate with tributaries of anterior abdominal veins, mainly epigastric vessels . LymphatBecause this entity is rare, the choice of surgical intervention is not clear. Historically, laparotomy was performed. Now, with advances of laparoscopic surgery, it is the safer and easier method for complete or partial excision of the falciform ligament. It also depends on the severity of the patient’s condition, the extent of disease, and the surgeon’s expertise. The operation performed in our patient was a subtotal resection of the falciform ligament, leaving only a small part of viable ligament in the umbilical fissure between segments III and IV. There has been only one report of a case in which a complete resection of the falciform ligament has been performed . AlthougInflammation and necrosis of the falciform and round ligaments is a very uncommon entity with limited literature available. However, some pearls can be gleaned from our case report and literature review. The mean age of presentation is 60 years old, and women and men are equally affected. Patients typically present with symptoms mimicking gallbladder disease, such as acute cholecystitis or symptomatic cholelithiasis. In cases of right upper quadrant or epigastric tenderness or peritonitis without positive findings of gallbladder inflammation or stones, or free fluid or gas, surgeons should be more suspicious of alternative pathology, such as round/falciform ligamentitis. CT is preferred to US to make the diagnosis. Surgical management is the definitive treatment, and a minimally invasive approach is preferred when feasible.