Background: Ewing sarcoma/primitive neuroectodermal tumor (ES/PNET) is a soft tissue malignancy arising from the neuroectoderm. While the locations of these extraskeletal manifestations are diverse, origin from the small bowel and small bowel mesentery is extremely rare. Intra-abdominal manifestations of ES/PNETs are nonspecific, and patients present with a wide range of symptoms, most frequently vague abdominal pain.Case Report: A 66-year-old female initially presented with vague and nonspecific symptoms of hypotension, anemia, dyspnea, and coffee-ground emesis. Imaging workup with computed tomography and fluorodeoxyglucose positron emission tomography demonstrated a metabolically active large mass involving the duodenum and measuring 10.3 × 8.8 × 12.3 cm. The mass was characterized as an ES/PNET on histopathologic diagnosis. The patient was treated with chemotherapy followed by radical resection and was disease-free at 1 year postpresentation.Conclusion: This case highlights that while ES/PNETs are rare tumors of the abdomen, they should be considered in cases of large soft tissue masses in patients presenting with nonspecific symptoms. To the best of our knowledge, this case is the fourth report in the literature of an ES/PNET involving the duodenum. When manifested in the musculoskeletal system, these tumors present with locoregional pain and a palpable mass. Advanced disease can also present with moderate fever, often accompanied by night sweats, and pathological fracture.2 Histopathologically, these tumors are composed of small round cells in a uniform distribution. These cells contain round nucleoli with fine stippled chromatin. The cytoplasm is scant, clear, or eosinophilic. Atypical features also include prominent nucleoli and irregular contours.3Initially described by Stout in 1918, Ewing sarcoma/primitive neuroectodermal tumor (ES/PNET) is a soft tissue malignancy arising from the neuroectoderm.4 While the locations of these extraskeletal manifestations are diverse, origin from the small bowel and small bowel mesentery is extremely rare. Intra-abdominal manifestations of ES/PNETs are nonspecific, and patients present with a wide range of symptoms, most frequently vague abdominal pain.5 We present the case of a 66-year-old female who presented with an ES/PNET involving the duodenum.Extraskeletal ES/PNETs occur in approximately 12% of patients with ES/PNET.2. At the time of initial presentation, the patient's medications included ramipril, metformin, and vitamin D supplement. Physical examination was notable for a small external hemorrhoid but was otherwise normal. Laboratory analysis at the initial ED visit demonstrated profound anemia with hemoglobin of 5.4 g/dL  and hematocrit of 18.3% . The patient was admitted, managed with 2 blood transfusions , and discharged home in stable condition the next day with a consultation placed for gastroenterology.A 66-year-old female initially presented to the emergency department (ED) for evaluation of hypotension, palpitations, and progressive dyspnea with exertion. The patient also reported palpitations and lightheadedness with positional changes for a period of 3 weeks prior to presentation, as well as a 6-month history of loss of appetite. Her medical history was notable for prediabetes, vitamin D deficiency, hyperlipidemia, and hypertension. Her surgical history was notable for cesarean section and tubal ligation. Patient's weight at presentation was 53.9 kg, and her body mass index was 22 kg/mThe patient returned to the ED 1 week later, presenting with new symptoms of coffee-ground emesis and abdominal pain. During the second ED visit, computed tomography (CT) of the abdomen showed a large heterogeneous mass measuring 10.3 × 8.8 × 12.3 cm  arising from the third segment of the duodenum , and 1C.CT-guided biopsy demonstrated a malignant round cell tumor with positivity for CD99, synaptophysin, CD56, and CD117, with negative expression of CD45, CD43, pancytokeratin, beta-catenin, chromogranin, WT1, desmin, S100, DOG1, and TLE1. Fluorescence in situ hybridization (FISH) analysis performed using DNA probes for EWSR1 demonstrated an unusual variant pattern of rearrangement of EWSR1 locus at 22q12 in 52% of nuclei, with 1 fusion and 1 free 5′ EWSR1 signal among 100 interphase nuclei examined.The patient's treatment course included palliative external beam radiotherapy (EBRT) to the mass at a dose of 20 Gy in 5 fractions that was initiated 17 days after her initial presentation and completed 23 days after her initial presentation. The EBRT resulted in the cessation of her emesis.The patient also completed 3 cycles of vincristine (2 mg intravenously [IV]), doxorubicin (113 mg IV), and cyclophosphamide  alternating with ifosfamide  and etoposide (160 mg IV) in alternating 14-day cycles initiated 3 weeks after her first presentation and completed 3½ months after her initial presentation. The patient then received treatment with 5 cycles of vincristine (2 mg IV), doxorubicin (113 mg IV), cyclophosphamide , and denosumab (60 mg subcutaneously) beginning approximately 3½ months after her initial presentation and completed 6 months after her initial presentation.Surgical course after systemic therapy included radical resection of the duodenal mass, open cholecystectomy, wedge gastrectomy, inferior mesenteric artery ligation, and small bowel resection approximately 7 months from initial presentation. The specimen from the radical resection  consiste6 A tumor differentiation score of 3 along with <50% necrosis and 24 mitoses/10 high power fields observed in this case assigns a grade 3 for the tumor . A single lymph node was identified and was negative for malignancy (0/1). Immunohistochemistry of the resection was positive for CD99 and negative for vimentin and nuclear WT1 expression  and a member of the ETS transcription factor family. The anatomic distribution of these tumors is wide, and case reports have described manifestations in the kidney, stomach, liver, and small bowel.escribed .11,13,144 Most patients are young, and patients >30 years often demonstrate tumor predilection to the soft tissues. While most cases are sporadic, germline mutations have been described. ES/PNETs are associated with FET-ETS fusion genes that encode chimeric transcription factors that regulate numerous genes, and an aberrancy in these transcription factors is thought to lead to the development of these tumors. The most common translocation, present in approximately 85% of cases, is t, which results in EWSR1-FL1 fusion transcript and protein.15ES/PNETs demonstrate a male to female ratio of 1.4:1.16 NKX2.2 positivity is more specific than CD99.17 Keratin expression is seen in 25% of cases.3Grossly, these tumors appear soft and grey-white with areas of necrosis. Histopathologically, ES/PNETs appear as uniform small round cells. These cells typically demonstrate round nuclei, poorly visualized nucleoli, and scant cytoplasm. Chemotherapy can lead to variable degrees of necrosis with replacement by loose connective tissue. CD99 positivity is seen in approximately 95% of ES/PNETs.4 Multimodal therapy for ES/PNETs has improved prognosis considerably. Response to neoadjuvant chemotherapy is a favorable prognostic factor.18The radiologic extraskeletal manifestations of ES/PNET are often nonspecific. Therefore, pathologic analysis is required for a definitive diagnosis. On imaging, intra-abdominal ES/PNETs can be misdiagnosed, with imaging features resembling gastrointestinal stromal tumors (GISTs), especially when they are present in the stomach and small bowel. Immunohistochemistry helps distinguish these tumors from GISTs, which are typically positive for CD117, DOG-1, and CD34.This report presents a rare location of the uncommon entity ES/PNET and demonstrates the often difficult and nonspecific clinical presentation of these neoplasms. While ES/PNETs are rare tumors of the abdomen, they should be considered in cases of large soft tissue masses in patients presenting with nonspecific symptoms. To the best of our knowledge, this case is the fourth report in the literature of an ES/PNET involving the duodenum.


Benign primary tumors are uncommon, with the majority of these tumors being leiomyomas; schwannomas of the esophagus are rare. Here, we present a case of a 78-year-old woman referred for complaints of intermittent dysphagia with a chest computed tomography scan showing a homogenous mass, compressing the esophagus. Upper gastrointestinal endoscopy revealed a submucosal mass, which was eventually diagnosed as a schwannoma after an endoscopic ultrasound with fine-needle aspiration and subsequent pathologic and immunohistochemical examination. Schwannomas could be managed conservatively. Schwannoma is a tumor of the peripheral nervous system. It originates in Schwann cells and usually refers to a benign, slow-growing tumor . While tA 78-year-old woman was referred for intermittent dysphagia for the past year. She denied any medical history and reported that dysphagia was present in both solids and liquids. She did not seek medical attention earlier due to the intermittent nature of her symptoms. She denied unintentional weight loss, hematemesis, dyspepsia, or reflux. Furthermore, she denied a family history of malignancy. Physical examination was unremarkable. A computed tomography scan of the chest showing a 3 cm, well-demarcated, enhancing, homogenous mass compressing the esophagus. Endoscopic evaluation showed a submucosal mass in the middle third of the esophagus with endoscopic ultrasound (EUS) demonstrating a 3 cm hypoechoic lesion with numerous anechoic spaces, consistent with cystic spaces Figures , 2.Fine-needle aspiration (FNA) demonstrated spindle cells surrounding acellular areas consistent with Verocay bodies on hematoxylin and eosin (H&E) stain Figure , and immSOX-10 and S-100 immunostaining markers showed positive results suggesting a neural origin of the tumor consistent with the diagnosis of esophageal schwannoma Figure .The patient’s preference was for conservative surveillance with the possibility of surgical intervention, if symptoms were to progress.Esophageal tumors are primarily malignant with benign esophageal neoplasms representing approximately 2% of all esophageal tumors . Due to While esophageal schwannomas are benign tumors, they show a hypermetabolic appearance on 18-fluorodeoxyglucose positron emission tomography, similar to findings of malignant esophageal tumors . Due to Once pathologic diagnosis is established, endoscopy +/- ultrasound for periodic surveillance has been suggested for asymptomatic lesions < 2cm in diameter, as these lesions often tend to be benign. Further interventions to evaluate and resect the lesion can be considered if any changes are encountered on surveillance. Endoscopic treatment may be attempted for small tumors, while enucleation by a thoracoscopic approach and esophagectomy may be considered for those lesions not amenable to endoscopic resection.Endoscopic ultrasound has been increasingly used to diagnose (when done in conjunction with FNA) and monitor esophageal tumors; it is the most accurate imaging modality to determine the locoregional spread of esophageal malignancy. Esophageal schwannoma is rare and should be considered in the differential when a submucosal esophageal mass is encountered. While complete excision of the mass for large lesions is widely performed, conservative management can be considered for minimally symptomatic patients, as in our case.


Bronchogenic cyst (BC), cyst lined by respiratory epithelium, is uncommon congenital anomaly of bronchial tree. Intraoral BC is extremely rare lesions. Here, we report the unusual presentation of 2-year-old boy with symptomatic cystic lesion at floor of month combined with ankyloglossia. The operation was performed under general anesthesia. Frenotomy was performed. Complete cystic removal was successful with minimal leakage of cyst wall. Sclerosing agent was injected at surgical site to prevent the residual undetected cystic malformation. Pathological examination was demonstrated a unicystic lesion lined by ciliated pseudostratified columnar and cuboidal cells. The final diagnosis was bronchogenic cyst. No postoperative complication was found. The long-term course was uneventful with no signs of recurrence at 1 year. To our best knowledge, a rare example of BC at the floor of mouth combined with ankyloglossia has never been reported. Bronchogenic cyst (BC), cyst lined by respiratory epithelium, is uncommon congenital anomaly of bronchial tree. They are usually located in the mediastinum near the tracheobronchial tree  or adjacent to the esophageal wall . IntrapeA 2-year-old boy presented with a slow growing mass under the tongue for 6 months. He was born without any anomaly. The development status was within average except delay speech due to the mass at floor of mouth. The physical examination revealed left sublingual cystic lesion measuring 1.0 × 1.0 cm. Ankyloglossia was found . CervicaThe operation was performed under general anesthesia. Frenotomy was performed. Complete cystic removal was successful with minimal leakage of cyst wall  and B. HBC, rare developmental anomalies, is lined by respiratory epithelium, i.e. ciliated, pseudostratified squamous epithelium, comprising mucus-secreting cells, smooth muscle cells and cartilaginous tissue. They are usually located in the mediastinum adjacent to the tracheobronchial tree or esophageal wall . CombineThe pathogenesis of BC has not yet been demonstrated, however, several hypotheses have been proposed in the literature. BC may result from an early aberrant nodule that becomes detached from the primitive tracheobronchial tree or may be derived from cells of the esophagotracheal ridge, forming an isolated bronchial structure .Mediastinal BC is often discovered at birth, usually asymptomatic and accidental discovered later in adult. When BC increases in size, it can be complicated by upper airway obstruction, causing feeding and even respiratory difficulties, infection and possibly abscess formation . HoweverOur patient presented a cystic lesion at floor of mount that interfered speech and breast-feeding. Moreover, it also associated tongue tie. The differential diagnosis of cystic lesion at the tongue include lymphoepithelial cyst, lingual thyroid, thyroglossal duct cyst, epidermoid cyst, dermoid cyst, mucocele, mucous retention cyst, cystic hygroma and hemangioma . The disInvestigation for accurate diagnosis is important. Computed tomography (CT) and/or MRI are performed. CT of BC typically shows sharply marginated mediastinal masses of soft-tissue or water attenuation. Sometime, CT can be confused with other lesions; MRI can be useful for elucidating the cystic nature of these lesions .MRI demonstrated multi-lobulated cystic mass, which showed hypersignal intensity on T2 weighted image, hyposignal intensity on T1 weighted image. Contrast to some literature that MRI revealed a homogeneous, unilocular cystic lesion with hypersignal intensity on T2 and hyposignal intensity on T1 with contrast enhancement after gadolinium infusion . AccordiMalignant transformation of BC to adenocarcinoma has been reported in adults with untreated chronic lingual cysts . HoweverSurgically enucleated is recommended for diagnosis, treatment and prevention of possible complications. Surgical enucleation should be performed at the time of the first complications or at the age of 1 year, constituting a balance between the risks related to general anesthesia , 15.Bronchogenic cyst at floor of mouth with ankyloglossia is an extremely rare anomaly. MRI should be the investigation in the presence of a cystic lesion of floor of the mouth in children in order to evaluate the precise anatomical relations, which helps to guide the diagnosis. Surgical excision is recommended. Bronchogenic cyst should be kept in mind in cystic lesion of floor of month.None declared.None.


Gossypiboma or textiloma denotes the formation of a mass lesion due to the development of foreign body reactions around the retained surgical item in the body. Most of the cases are asymptomatic in the initial postoperative period. Due to the dense adhesions, most of the cases of intra-abdominal gossypiboma are treated by an open approach. Here we present a case of a 38-year-old lady with left iliac fossa pain one month after caesarean section. Contrast-enhanced computed tomography of the abdomen revealed gossypiboma. The patient was successfully treated with laparoscopic removal of the gossypiboma. The term gossypiboma is used to describe surgical sponges or gauze retained inside a patient’s body after a surgical procedure -4. The bThis unintentional but avoidable surgical complication causes serious damage to the patient’s health. The current estimate of retained surgical items is one in 10,000, with surgical sponges accounting for the majority of cases. However, the exact incidence is not known as it is under-reported due to the fear of medicolegal consequences. The most common sites of gossypiboma are the abdomen 56%), pelvis (18%), and thorax (11%) [6%, pelviThe foreign body should be removed once diagnosed as it can erode in the adjoining structures leading to organ damage and fistula formation -8. Here,A 38-year-old female presented with left lower quadrant pain for seven days associated with low-grade fever. There was no history of vomiting, altered bowel habits, hematemesis, melena, or weight loss. She had undergone a lower segment caesarean section one month back. On clinical examination, she was febrile and had mild tenderness in the left lower quadrant of the abdomen. The previous Pfannenstiel incision of the caesarean section had completely healed with no palpable lump or cough impulse. Routine blood investigations were within normal limits. Abdominal ultrasonography revealed a bulky uterus with a 42 cc collection in the anterior abdominal wall at the left lateral end of the Pfannenstiel scar. There was an irregular-shaped large echogenic mass of 109 x 77 mm with posterior acoustic shadow in the left iliac fossa raising the suspicion of gossypiboma. In contrast-enhanced computed tomography (CECT) of the abdomen, a large well-defined peripherally enhancing hypodense lesion measuring 12 x 11 x 9 cm with multiple small air foci were noted in the left iliac fossa causing mass effect and compression of the adjoining small and large bowel loops suggestive of gossypiboma (Figure Pneumoperitoneum was created by Veress needle through Palmer’s point. The 10 mm camera port was placed in the right upper quadrant, and two 5 mm working ports were placed in the right iliac fossa and epigastrium, respectively. On diagnostic laparoscopy, the small and large bowel loops densely adhered to the anterior abdominal wall in the left iliac fossa. Adhesiolysis was done, and the cavity was opened. About 100 ml of seropurulent fluid was drained. A retained surgical sponge was seen densely adhered to the cavity lined by granulation tissue Figure .With sharp and blunt dissection, the surgical sponge was separated from the wall of the cavity. Thorough lavage was given. No leakage of bile or enteric content from the wall of the cavity was noted. A tube drain was placed in the cavity, and the retained surgical sponge was extracted through a small lower midline incision. The operative time was 120 minutes with a blood loss of 50 ml. The postoperative course was uneventful, with a hospital stay of five days. The drain was removed on the fifth postoperative day before discharge as the output was minimal. Pus culture was sterile. On the last follow up at three months after surgery, the patient was doing well without any complications.Gossypiboma is derived from the words “Gossypium” and “boma”, which means ‘cotton wool’ and ‘to coverup’, respectively. The first case was described by Wilson et al. in 1884. The incidence of gossypiboma varies from one in 8801 to one in 18760 of inpatient operations . HoweverMost of the patients are asymptomatic in the early postoperative period. However, the retained surgical items can erode the surrounding structures and lead to fistula formation with the gastrointestinal and genitourinary tract . Once thIn the immediate postoperative period, a plain radiograph may show the radio-opaque line present in the sponges . HoweverDuring surgical exploration, the gossypiboma is usually covered by the surrounding structures, with dense adhesions as seen in the present case. In long-standing cases, there may be partial or complete migration of the retained surgical sponge into the adjoining hollow viscus -8. The fA retained sponge is associated with significant morbidities. Hence, a systematic approach during the operation is essential to reduce its incidence. The first step is to perform sponge counts multiple times during surgery. The sponge count must be performed prior to the start of any procedure, at the time of addition of a new sponge, prior to the closure of any cavity, and at the time of closure of the incision. If any discrepancy is found, the surgical team should look for the missing item . The AmeGossypiboma is an avoidable surgical complication. Intraoperative gauze counting and double-checking with the surgical team is crucial to facilitate early recognition and detection of any missed sponge or instrument to decrease its occurrence. Gossypiboma can have serious consequences on the patients’ health and lead to medicolegal challenges for the treating surgeons. Once detected, it should be surgically removed. Laparoscopy can be an excellent approach for the retrieval of gossypiboma in appropriately selected patients.


Surgical resection of gallbladder cancer with negative margins is the only potentially curative therapy. Most patients with gallbladder cancer are diagnosed in an advanced stage and, despite the availability of several chemotherapies, the prognosis remains dismal. We report a case of locally advanced gallbladder cancer that was successfully treated with effective cisplatin plus gemcitabine, followed by curative resection.A 55-year-old Japanese female was hospitalized with right hypochondrial pain. Enhanced computed tomography revealed a 49 × 47 mm mass at the neck of the gallbladder, with suspected invasion of the liver and right hepatic artery. Endoscopic retrograde cholangiopancreatography demonstrated displacement of the upper bile duct. Intraductal ultrasonography showed irregular wall thickening and disappearance of the wall structure in bile ducts from the B4 branch to distal B2 and B3. Percutaneous transhepatic biliary biopsy revealed a poorly differentiated carcinoma. The patient was diagnosed with unresectable gallbladder cancer (T4N0M0 stage IVA). Cisplatin plus gemcitabine chemotherapy was initiated. After six courses of chemotherapy, enhanced computed tomography showed that the mass in the neck of the gallbladder had shrunk to 30 mm, Endoscopic retrograde cholangiopancreatography showed improvement of the hilar duct stenosis. A biopsy of the bile duct mucosa showed no malignant cells in the branch of the left and right hepatic ducts, the left hepatic duct, or the intrapancreatic ducts. The patient underwent conversion surgery with right and segment 4a liver resection, extrahepatic duct resection, and cholangiojejunostomy. The histopathologic diagnosis showed that the tumor cells had shrunk to 2 × 1 mm, and that R0 resection of the T2aN0M0 stage IIA tumor was successful.Although conversion surgery for gallbladder cancer is rarely possible, curative resection may offer a better prognosis, and it is important to regularly pursue possibilities for surgical resection even during chemotherapy. Gallbladder cancer (GBC) is a rare disease accounting for 4% of all gastrointestinal malignancies, but there is a higher incidence in countries such as India, Japan, Chile, and Mexico , 2. Due Chemotherapy is the standard treatment for unresectable gallbladder cancer. Combination chemotherapies with gemcitabine and cisplatin (GC) or gemcitabine and S-1 (GS) are recommended as first-line chemotherapy regimens for nonresectable biliary tract cancer. Combination chemotherapy with gemcitabine, cisplatin, and S-1 (GCS) is also a candidate regimen . On the Herein, we report a case of locally advanced GBC that was successfully treated with effective cisplatin plus gemcitabine followed by curative resection.A 55-year-old Japanese female was hospitalized with right hypochondrial pain. Laboratory tests showed elevated hepatobiliary enzymes and C-reactive protein. Serum levels of carcinoembryonic antigen and carbohydrate antigen 19-9 were 0.8 ng/ml and 3.2 U/ml, respectively. Enhanced computed tomography (eCT) imaging revealed a 49 × 47 mm mass at the neck of the gallbladder, with suspected invasion of the liver and right hepatic artery  or gemcitabine and S-1 (GS) are recommended as first-line chemotherapy regimens for nonresectable biliary tract cancer. Combination chemotherapy with gemcitabine, cisplatin, and S-1 (GCS) is also a candidate regimen . In thisSince gemcitabine or S-1 have been administered to GBC, there have been case reports of conversion surgery. Based on a review of the PubMed database using the key words “gallbladder cancer,” “chemotherapy,” and “resection,” we found six cases of conversion therapy for initially unresectable locally advanced gallbladder cancer, including our case (Table et al. [Recently, neoadjuvant chemotherapy has been proposed, knowing the potential for an increase in the resectability rate and overall survival. Hakeem et al.  reportedIn our case, eCT showed a marked reduction of the mass after six courses of GC therapy. An intrabile duct biopsy was performed by ERCP to rule out bile duct extension. The presence or absence of invasion to the right hepatic artery and hepatic invasion could not be evaluated. The liver remnant function was good and curative resection was considered possible by right and segment 4a liver resection and extrahepatic bile duct resection; thus, conversion surgery was performed. In the resected specimen, the tumor showed a marked shrinkage of approximately 2 mm. Residual tumor cells were found in a small part of the gallbladder neck and gallbladder wall at the hilum of the liver. No hepatic invasion, extrahepatic bile duct extension, or right hepatic artery invasion was observed. Limited surgery, such as liver segment 4a + 5 resection and extended cholecystectomy might have been possible while performing immediate frozen-section analysis, but the fibrosis that reflected the disappearance of tumor cells made it difficult to ablate the right hepatic artery. In GBC cases with suspected vascular invasion before chemotherapy, though chemotherapy is effective, surgery after chemotherapy may require major surgery.This is a case of advanced GBC with a pathologically proven remarkable response to GC therapy.This case is of a locally advanced GBC with marked tumor shrinkage with GC therapy. There are no reports that achieve a complete response from chemotherapy alone. Therefore, curative resection following GC therapy was important for the patient. There are few reports on conversion surgery for unresectable GB; therefore, the optimal chemotherapy regimen, preoperative period, and indications for curative surgery optimal surgical techniques are not clear. The short-term results, such as R0 resection rate and safety, and long-term results are also unclear. Further accumulation of cases is necessary.Although conversion surgery for GBC is rarely possible, curative resection may offer a better prognosis, and it is important to regularly pursue possibilities for surgical resection even during chemotherapy.


We report a 40-year-old man, with an unremarkable personal and family history, who presented for evaluation of an asymptomatic papule located on his right cheek. Histopathology revealed an encapsulated neoplasm within the dermis; composed by narrow, elongated, and wavy cells with an ill-defined cytoplasm, dense chromatin and tapered ends interspersed with collagen fibers. Pathologic findings were consistent with tissue of Antoni B pattern. The diagnosis was an infraorbital schwannoma. The incidental finding of rare tumors like this, should make clinicians consider a greater spectrum of differential diagnoses for a unilateral skin-colored papule on the cheek of patients. A 40-year-old male, with an unremarkable personal and family history, presented for evaluation of an asymptomatic papule located on his right cheek that had been present since childhood. He had not received any treatment. Physical examination revealed a solitary, circumscribed, firm, skin-colored, 4–6 mm papule A. DermosHistopathology  A and B a)Dermal nevusb)Sebaceous hyperplasiac)Infraorbital schwannomad)TrichoepitheliomaSchwannomas, also called neurilemmomas, are benign ectodermal neoplasms originated from peripheral, cranial, or autonomic nerve Schwann cell sheaths.None declared.Irving Llibrán Reyna-Rodríguez: Study conception and planning; preparation and writing of the manuscript; data collection, analysis, and interpretation; critical literature review.Sonia Chavez-Alvarez: Study conception and planning; critical literature review; effective participation in research orientation; approval of the final version of the manuscript.Jorge Ocampo-Candiani: Study conception and planning; critical literature review; effective participation in research orientation; approval of the final version of the manuscript.None declared.


Although inguinal bladder hernia associated with obstructive uropathy is an extremely rare entity, it should be suspected in elderly patients with bladder outlet obstruction presented with inguinal hernia and lower urinary tract symptoms. Although inguinal bladder hernia associated with obstructive uropathy is an extremely rare entity, it should be suspected in elderly patients with bladder outlet obstruction presented with inguinal hernia and lower urinary tract symptoms. A 88‐year‐old male with history of severe benign prostatic hyperplasia with underactive bladder under permanent Foley catheter due to poor response to TURP presented with symptoms of catheter‐related urinary tract infection. Physical examination revealed right incarcerated inguinoscrotal hernia with the right testicle within the scrotum separable from the hernia and without findings of bowel obstruction. Kidney function tests suggested stage‐III acute kidney injury (serum creatinine 4.8 mg/dl with a 3.5 times increase from baseline). Renal ultrasonography revealed moderate bilateral hydronephroureterosis. CT performed to establish diagnosis Figure .1Bladder apex, body, and fundus (blue arrow) along with the trigone (red arrow) protruded into the hernia sac without herniation of bowel loops, as shown in Figure Inguinal bladder hernia is rare with an incidence of 1%–3% of all inguinal hernias. Pathophysiology involves urinary bladder outlet obstruction leading to chronic distention and atony along with abdominal wall weakness.None declared.All authors equally accessed the data and contributed to the preparation of the manuscript. BKA and HA were equally responsible for making and performing treatment decisions. HA reviewed the manuscript for critical intellectual content and had the final approval.The present article does not contain any studies with human or animal subjects performed by any of the authors.Informed consent was obtained from the patient.


Plexiform fibromyxoma (PF) is a rare mesenchymal neoplasm which can be misdiagnosed as the gastrointestinal stromal tumor. This tumor almost formed a lobulated intramural/submucosal mass in the gastric antrum and prepyloric area. It was considered as a benign tumor that exhibited no recurrence, metastasis, or tumor-related mortality. In this study, we reported 2 cases of gastric PF. The first case was a PF patient coexisting with gastric adenocarcinoma. The second case occurred in the gastric upper body close to gastric fundus. They underwent distal gastrectomy and laparoscopic partial gastric resection, respectively. Both of them exhibited a plexiform growth pattern in the submucosa, muscularis propria, and subserosal adipose tissues. The nodules were composed of abundant myxoid or fibromyxoid matrix riching in small thin-walled blood vessels and bland-looking spindle cells. The first case partially showed staggered growth pattern of PF and adenocarcinoma. Immunohistochemically, the spindle cells were diffusely immunoreactive for SMA and vimentin, and focally immunoreactive for CD10. It was important to distinguish the PF from other spindle cell tumors involving the stomach. Most PF is arisen from the antrum and pyloric region, forming a lobulated intramural/submucosal mass. In clinical settings, PF is often misdiagnosed as GIST due to similar clinical manifestations. Rare cases showed synchronous occurrence of PF and other primary gastrointestinal tumors. In this study, we reported 2 cases with PF in stomach, together with describing the clinical characteristics, histopathologic and immunophenotypical features, as well as the discussion on the misleading differential diagnosis. More importantly, among the 2 cases, 1 showed co-existence of PF and gastric adenocarcinoma that had never reported in the previous literatures.Plexiform fibromyxoma (PF), also known as plexiform angiomyxoidmyofibroblastic tumor, is a rare benign mesenchymal neoplasm of the stomach. It was first described in 200722.1A 64-year-old female patient presented to our department due to epigastric discomfort for 7 days. Laboratory findings were normal. Computed tomography (CT) scan and endoscopic ultrasound demonstrated a solid mass within the submucosa and muscularis propria of the gastric antrum. Endoscopic biopsy showed epithelial dysplasia in focal mucosa, and possibility of gastric adenocarcinoma should not be excluded. On this basis, distal gastrectomy was conducted. Grossly, the mass (2.5 cm × 2 cm × 1.7 cm) was localized at the antrum close to the pylorus region with a mucosa ulceration (1 cm × 1 cm). The tumor mass was well demarcated with a solid nature and mucoid appearance. Its growth was in a multinodular pattern, mainly involving the submucosa, muscularis propria, and subserosal adipose tissues.For the histopathologic findings, 2 different morphological manifestations were observed as follows: The dysplasia glands infiltrated the mucosa to muscularis propria. On this basis, moderately differentiated adenocarcinoma was defined Fig. A. A plex2.2A 39-year-old male patient was admitted to our hospital for routine gastroscopy, which revealed an elevated mucosal lesion (2.0 cm × 2.0 cm) with ulceration in the gastric upper body. CT scan showed a mass within the wall of the gastric upper body close to gastric fundus. Laboratory findings were within the normal ranges. Physical examination on the abdomen was unremarkable. Endoscopic biopsy showed no malignant cells and endoscopic ultrasound-guided fine-needle aspiration biopsy showed bland-looking spindle cells. On this basis, partial gastric resection was performed under laparoscopic assistance as he was suspected with GIST.A multinodular growth pattern was noticed mainly involving the submucosa, muscularis propria, and subserosal adipose tissue. The tumor was well demarcated and the cut surface was solid and mucoid Fig. A. Histol3 first described 2 cases with plexiform angiomyxoidmyofibroblastic tumor in 2007. In 2009, Miettinen et al described 12 patients with PF. In 2010, this unique gastric mesenchymal tumor was defined as plexiformfibromyxoma by World Health Organization. Since then, more cases were reported all over the world.–11PF is a rare gastric mesenchymal malignancy that has been reported among more than 100 cases. Takahashi et al,13 As is known to all, stomach is the most common location for GIST with coexisting tumors and the major types of GIST-associated cancers are gastrointestinal carcinomas (47%). Due to rarity of the disease with tumors of different histotypes in the same organ, it is still not clear whether the synchronicity is accidental or related pathophysiological processes. Genetic mutations may be the basis for tumor predisposition in patients with 2 synchronous gastric tumors. In most cases with synchronous tumors, they presented separate lesions in different locations. In our case, an isolated lesion was noticed in which PF and adenocarcinoma showed staggered growth pattern causing challenges in the diagnosis.In this study, we reported 2 cases of PF in stomach, among which 1 showed coexistence of gastric adenocarcinoma. To our best knowledge, there was no such case before. Meanwhile, rare cases with simultaneous development of GIST and adenocarcinoma are available. PF was commonly reported in adults and some pediatric patients,17 with similar pathological features and benign clinical behaviors, except increased tumor growth in pediatric PF. This suggested that PF was the same disease entity regardless of age of onset in the previous description. The size of the tumors ranged from 0.8 cm to 17 cm in the maximal diameter (median: 4.0 cm). Although most PF is located in the gastric antrum and prepyloric area, the tumor could originate from the gastric body, duodenum, the jejunum, and the colon.,19–21The age of PF patients was in a range of 5 to 81 years with no gender differences.,11 The microscopic features of PF were relatively unique. These tumors often presented a multinodular growth pattern, involving the intra-mucosal to serosal of the stomach. The tumor nodules contained a prominent myxoid stroma rich in small thin-walled blood vessels with low to middle cellularity composed of bland spindle without cytological atypia, mitotic figures, or necrosis. In most areas, the tumor cells were arranged in a loose manner. The extracellular matrix within these tumors was usually positive by Alcian-blue staining. In some nodules, the collagenous stroma was densely hyalinized. Frequent ulceration, mucosal invasion, and vascular invasion had no adverse significance in these tumors. Immunohistochemically, the tumor cells were diffusely immunoreactive for SMA and vimentin, and focally immunoreactive for H-caldesmon, desmin, and CD10. The expression of myogenic markers may suggest that there were tumor cells with myofibroblastic-fibroblastic phenotype and true smooth muscle differentiation.PF patients typically present with abdominal discomfort and pain, anemia, and upper gastrointestinal bleeding, which are mainly caused by mucosal ulceration or other non-specific upper digestive tract symptoms. The diagnosis of PF is highly depending on pathological and immunochemical examinations, as the CT, magnetic resonance imaging, and gross examination findings of PF are similar with GIST. Mucosa ulcerations occurred in nearly half of the cases, which led to gastrointestinal hemorrhage and secondary anemia. GIST should be excluded initially. Few GISTs exhibited a plexiform and myxoid appearance similar to PF. However, GISTs typically expressed CD117, DOG-1, and CD34 and were negative for SMA, S-100, desmin, and AE1/AE3. In addition, 80% and 5% to 8% of GIST were primarily driven by the mutations in KIT and PDGFRA, respectively. These aspects were not seen in PF.The differential diagnosis of PF includes other spindle cell tumor involving the stomach. The tumor cells were negative for CD117, DOG1, CD34, S100, β-catenin, STAT-6, and ALK that played important roles in differential diagnosis. As the most common gastric mesenchymal tumors, About 50% of cases harbored rearrangements of ALK gene. Fibromatosis usually arises in mesentery but less gastric involvement has been reported. It has an infiltrative growth pattern and comprises of myofibroblasts. In focal parts, there were loose storiform structures. About 75% to 90% of cases exhibited nuclear expression of β-catenin and SMA immunoexpression.Myxoid leiomyoma, usually involving the esophagus, typically shows numerous spindle cells with cigar-shaped nuclei and eosinophilic cytoplasm. It contained no multinodular pattern, but was commonly seen in PF patients. Moreover, gastric leiomyoma was immunoreactive for desmin, actin, calponin, and h-caldesmon. Gastrointestinal schwannoma often formed a nodule lymphoid cuff with infiltrative margin and lymphoid cuff. They present diffuse immunoexpression of S-100 protein and SOX10. Inflammatory fibroid polyp occurred in a submucosal location. It was composed of bland short spindled cells which often arranged concentrically around vessels. There were many eosinophilic granulocytes in the loose fibromyxoid stroma. Most inflammatory fibroid polyp expressed CD34 and SMA rather than CD117, DOG-1, and S100. Inflammatory myofibroblastic tumor was composed of spindle myofibroblasts with lymphocytes and plasma cells in the background with intermediate biological behavior. In some cases, there might be occurrence of myxoid, vascular, and inflammatory areas resembling nodular fasciitis.GLI1 activation within the Hh pathway, including 2 (12.5%) with GLI1 amplification and 3 (19.75%) with MALAT1-GLI1 oncogenic fusions. Banerjee et al reported the first report of recurrent PTCH1 loss in PF based on next generation sequencing and proposed that targeted Hh pathway inhibition with SMO antagonists might represent a target for treating a subset of PF. However, no KIT mutations or PDGFRA mutations are identified in PF.To date, only few reports have investigated the molecular biology of PF. In the previous study, 5 cases (31.25%) showed  laparoscopic endoscopic cooperative surgery can be appropriate in the diagnose and therapy of submucosal tumors. It can prevent the deformation of the stomach, especially the tumor is located near the pylorus. PF acts as a benign tumor with few cases showing recurrence or distant metastases after complete surgical resection. In this study, the prognosis of case 1 was depending on the adenocarcinoma factors as PF acted as a benign neoplasm. In future, further studies are required to investigate the potential causal relationship between these synchronous tumors.Nowadays, the distal gastrectomy or partial gastrectomy is the main treatment option for PF involving the stomach. In a previous study,In summary, PF is a rare mesenchymal tumor mainly occurring in the gastric antrum, which is mainly diagnosed based on histological and immunohistochemical findings. Most of PF patients showed a lobulated submucosal mass and were often misdiagnosed as GIST because of the similar clinical manifestations. In clinical practice, more attention should be paid to it as it can coexist with gastric adenocarcinoma, resulting in a poor prognosis in the patients.Conceptualization: Lu Song.Data curation: Lu Song.Formal analysis: Lu Song.Funding acquisition: Shuming Qin.Investigation: Shuming Qin.Methodology: Shuming Qin.Software: Daosheng Li.Supervision: Daosheng Li, Gang Hou.Validation: Daosheng Li, Gang Hou.Visualization: Gang Hou.Writing – original draft: Min Lin.Writing – review & editing: Xiaomei Li.


We report a case of prune perineum syndrome, an extremely rare entity with only four cases reported to date, describing some typical clinical and radiologic features. We also present a newly associated imaging finding, the terminal ventricle’s cystic dilatation, and briefly discuss the differential diagnosis. Noteworthy, only in the two most recent reported cases, the patient reached childhood. PPS is characterized by both sacrococcygeal spine and perineal malformation, so as absent or hypoplastic genitalia, being the most typical finding a large “prune-like” perineal mass, giving its known definition. This report aims not only to describe the fifth case of PPS, but a newly associated feature, the cystic dilatation of the terminal ventricle.Prune perineum syndrome (PPS) is a poorly recognized clinical entity. Only four cases have been published in the literature, the first being described in 1979 by Peeden et alThe patient was born from a healthy first-time mother, with no familial history of congenital malformation, at the 37th week of a twin pregnancy, being the 2nd twin of cesarean delivery. Her birth weight was 2,705 g, cephalic perimeter 36 cm, thoracic perimeter 30 cm, length 45 cm, 1st- and 5th-minute Apgar 9 and 10, respectively. Mother and newborn blood types were B positive.The most evident clinical finding was a large mass extending from both gluteal regions and perineum, with a tiny central orifice draining urine, without a definitive urethral meatus. No external genitals or anus were characterized. A dimple was noted on her sacral region, a few centimeters below the iliac crest line, and a bony appearance elevation was seen during spine palpation . There wThe other twin was born without any clinical findings or evident malformations and remained normal cognitive and growing development. There are no other siblings.Due to the entity’s unrecognition, the newborn was observed and investigated during the first few days. The karyotype was normal (46 XX) – so as her twin sister, the echocardiogram showed a patent foramen ovale and persistence of the left superior vena cava. Spine ultrasound detected a rhombus conus medullaris termination with no masses within the spinal cord. Abdominal ultrasound detected an anterior rotation of the left kidney. No bladder or uterus has been detected. Spine radiography showed a pubic diastasis and sacrococcygeal eversion, with a bony protrusion oriented upward.Abdominal MRI was performed in her 2nd-day hospitalization , showingDue to progressive abdominal dilatation and colonic obstruction caused by anus atresia, a left colostomy was performed on her 5th day of hospitalization. The patient required 2 days of mechanical ventilatory post-operative assistance and has been discharged on her 8th-day, without complications. No abnormalities were found in brain MRI.Spine MRI findings were consistent with developmental anomalies characterized by the anterior fusion of the L4 and L5 vertebral bodies (block vertebra), thoracolumbar scoliosis with right convexity, rectification, and posterior orientation of the sacrococcygeal elements towards the skin/subcutaneous cellular tissue , where a fibrous band, part of the external “dimple” was inserted, without communication to the thecal sac .Additionally, there was a cystic dilatation of the terminal ventricle at the level of the distal portion of the medullary cone (T12-L1), measuring approximately 8.0 mm in its largest diameter. A small lipoma of the filum terminale was also noted .Currently, the patient has 6 months of age with normal neurological and psychomotor development and remains with a colostomy bag, so as under intermittent catheterization. The next surgical step aims for urinary diversion through a continent abdominal reservoir and “prune-like” mass correction. Although pediatric surgeons have discussed this strategy, due to lack of literature evidence, it remains undefined. Due to pandemics, genetic analysis to reveal possible mutations were not done yet.PPS is an extremely rare and unknown entity. A typical “key” diagnostic feature of PPS is the presence of a “prune-like” redundant perineal mass with mucous  and cutaneous  epithelium containing malformed pelvic structures with a common fistulous ureteral pathway draining into the perineal cavity. To date, there is no connection between this entity and sex because all previous related four cases were 46 XY, differing from ours. Twin pregnancy maybe is not a risk factor either, explained by the fact that our case is the only one published in this presentation.3 postulated an embryologic explanation for the orifice draining urine in the mass, which consists of a prenatal probable fistula rupture between the “perineal bladder” and skin before due to increased intravesical pressure. Still, no other explanations were found in the literature. As in our case, all the described patients with PPS have normal motor and cognitive development.Lopes et alMisdiagnosis can be made with associated anorectal and genital tract malformations, like anal atresia-persistent cloaca, chromosomic aberrations related to anal atresia, maternal diabetes mellitus associated sacral dysgenesis, and progesterone-estrogen or progesterone hormonal pills anal–rectal-related malformations, some of which included in the VACTERL spectrum.5,6 Major differences are syringohydromyelia and hydromyelia.Terminal ventricle cyst or fifth ventricle is a fusiform ventricle, measuring 8–10 mm in length and 2–4 mm in transverse diameter located in the conus medullaris. A few relations between this finding and congenital abnormalities have been postulated, but it is currently known for its typical benign course3 The fourth case provided relevant information and strategies aiming at malformations correction and life quality improvement, consisting first in an osteotomy, followed by definitive colostomy, cystoscopy evaluation, cystoplasty, perineal and abdominal correction with mesh, bilateral orchiopexy, treatment of the vesicocolonic fistula, sigmoidectomy and appendivesicostomy.4 The cited patient is 6 years old, walks with no assistance, and has good abdominal tonus.Four cases of PPS have been described to date with no consensus in therapeutic strategies. The third case was first managed by doing a continent cutaneous ileal neobladder. However, 4 months after surgery, the patient developed an acute obstructive abdomen, followed by septic shock and death.PPS is an extremely rare and underrecognized entity.PPS’s key diagnostic feature is the presence of a “prune-like” redundant perineal mass containing malformed pelvic structures with a common fistulous ureteral pathway draining into the perineal cavity.The main differential diagnosis of this entity consists of VACTERL disease spectrum.Cystic dilatation of the terminal ventricle is a new associated finding in this entity.There is still no consensus in the therapeutic management of PPS, and the prognosis remains low. Still, some new strategies have been published to aim malformations correction and life quality improvement.PPS is an extremely rare disease, and its correct recognition is vital to avoid misdiagnosis with other malformations, with emphasis on VACTERL. Our case is the fifth in the literature and the first with an associated cystic dilation of the terminal ventricle. Due to lack of experience and possible underrecognition of this entity, we strongly suggest future research focusing on both physiopathological mechanisms and associations, as well as patient’s management. Although cystic dilatation of the terminal ventricle could be an incidental finding on the neonatal lumbar spine, it can be present in this syndrome and may add in future reports and identification of this condition.


A ruptured Baker’s cyst is a rare presentation and may mimic deep vein thrombosis (DVT) or acute thrombophlebitis. In rare cases, it may present with infection or compartment syndrome. We present our experience related to a case of a ruptured Baker’s cyst and its management. A 54-year-old female presented to us with knee pain, which was initially managed conservatively. After six weeks, she came to us with severe pain and swelling in her left calf and foot. It was an acute presentation and DVT was suspected initially. Ultrasound color Doppler showed no DVT and then MRI revealed it to be a ruptured Baker’s cyst. The patient was subsequently managed conservatively and her condition improved in 12 weeks of follow-up. A high index of suspicion and knowledge is required to diagnose a ruptured Baker’s cyst, and most of the patients respond well to conservative management. Baker’s cyst, also known as a popliteal cyst, is formed due to fluid accumulation in the pre-existing bursae at popliteal fossa . The mosIn this report, we present a case of a ruptured Baker’s cyst and discuss its clinical presentation, diagnosis, and our experience in managing the patient.A 54-year-old female presented to us with a complaint of left knee pain for six months. On examination, the knee range of motion was full, with no ligamentous laxity. There was tenderness at the femoral attachment of the lateral collateral ligament (LCL) and also at pes anserinus. Also, a Baker’s cyst was present at the popliteal fossa. Local infiltration with an injection of bupivacaine and methylprednisolone acetate was done, and the patient was educated about quadriceps- and hamstring-strengthening exercises. The patient reported good relief after the procedure.After 1.5 months, she presented with severe swelling in the left lower limb extending from knee to foot. There was no history of any trauma and no signs of infection. On physical examination, swelling could be appreciated in the left leg and foot. The swelling was more prominent at the dorsal aspect of the leg. There was tenderness at the middle one-third of the calf. The dorsiflexion of the foot seemed to intensify the pain (Homans sign). The plantarflexion power of the foot was normal. Hence, we suspected DVT and got an ultrasound color Doppler of the patient. The color doppler showed no signs of DVT.Then, on careful questioning, she gave a history of pain exacerbation while doing vigorous exercises of quadriceps and hamstring. Also, she stated that she had noticed some fluid going down the leg. This prompted us to suspect a ruptured Baker’s cyst, and an MRI of the leg was performed. The MRI showed a fluid collection at the intermuscular plane, which correlated with the site of tenderness at the calf Figure , 2, 3, 4The patient was managed conservatively and kept on regular follow-up. On subsequent follow-ups at two weeks, six weeks, and 12 weeks, the swelling completely subsided and the patient became pain-free.Baker’s cyst is one of the most common cystic lesions of the knee joint. It is formed by the distension of gastrocnemio-semimembranosus bursa. Unlike other bursae of the knee joint, this bursa communicates with the cavity of the knee joint . The incThe presentations in cases of ruptured Baker’s cyst closely resemble those of DVT . It coulUltrasound is the initial investigation of choice as it rules out DVT and also indicates the possibility of a ruptured Baker’s cyst. The collection of fluid in the intermuscular plane points to a ruptured Baker’s cyst. Although a few authors consider ultrasound as the key diagnostic tool , in our The ruptured Baker’s cyst usually presents with acute pain and swelling. Hence, the only treatment required is pain management with anti-inflammatory medications and local heat . SurgicaThe ruptured Baker’s cyst is a rare entity and is often missed on initial presentation as its symptoms closely resemble those of DVT or acute thrombophlebitis. Though most of the patients respond well to conservative management, a high index of suspicion is needed to make an early diagnosis.


Peripheral ossifying fibroma (POF) is a benign localized lesion originating from gingival and alveolar oral mucosa. Its origin can be cells of periodontal ligament. The lesions usually develop in women in their twenties. POF is a complex clinical and histological diagnosis due to its shared characteristics with many other conditions. In this paper, we presented a case of an atypical peripheral ossifying fibroma (POF) in the left lateral part of the mandible in a 70-year-old male patient who had two semicircular bridges supported on four implants in the upper and lower jaws. A review of CBCT and orthopedic imaging showed no visible intraosseous changes. Histological analysis revealed the diagnosis of POF. The case in question is interesting, as elaborated on in the discussion section of this paper because POF is usually found in female patients aged between 20 and 30 years. Peripheral ossifying fibroma (POF) is a non-malignant localized lesion originating from gingival and alveolar oral mucosa, clinically manifesting as a painless, slow-growing, hard nodule, usually smaller than 2 cm. It is histologically characterized by a fibrous tissue affected by a variable number of fibroblastic cells. The presence of well-defined islands of metaplastic bone can be extremely helpful when setting a differential diagnosis ,2. VarioA male 70-year-old patient came to the Department of Oral Surgery, University Hospital Centre Zagreb with a voluminous fibrous mass in the distal region of the left mandible . The patThe mass was then sent for pathohistological (PHD) analysis, which showed that the mass has a reactively changed multilayered epithelium with abundant mononuclear linear inflammatory infiltrate underneath. There were no elements of Lichen. Fragments resembling cemento-osseous lacunae were found inside the thick fiber stroma. A restricted area with clusters of hemosiderin and gigantocellular cells was found in the middle of the sample. Interestingly, a small salivary gland with multiplied intra- and interlobular tissue with widened performing ducts was found in the sample. The outer edges of the sample also contained part of the mucocele wall .Poorly coordinated intermaxillary relations between the upper and lower acrylic bridge, as well as parafunctional movements, can be considered distinct predisposing factors for the development of this formation.The appearance of POF is of unknown etiology and can be linked to different susceptibility factors such as bruxism, dental plaque and inadequately set or adjusted prosthetic teeth. Different names for POF are used in the literature, such as fibrous epulis or calcifying fibroblastic granuloma . PeripheIntraoral ossifying fibroma has been described in the literature since the late 1940s. Various names were given to comparable lesions, such as epulis, peripheral fibroma with calcification, peripheral fibroma with osteogenesis, calcified fibroblastic granuloma, peripheral cementifying fibroma, peripheral fibroma with cementogenesis and peripheral cemento-ossifying fibroma ,12. ArouOgbureke et al. presented a case of a 44-year-old male who came into the emergency room and complained about swelling in the back segment of the right mandible that had been going on for three months. His family history included diabetes mellitus type 2, cardiovascular diseases and hypertension. He had two implants in the right distal quadrant near the lesion three months before the appearance of the mass. It was concluded that it was quite hard to clinically differentiate peripheral gigantocellular lesion and peripheral ossifying fibroma. The difference can be established histologically. Gigantocellular cells are present in both POF and peripheral gigantocellular granuloma, whereas cemento-ossifying lacunae is only present in POF .®, Foothill Ranch, CA, USA) was used to remove the mass. Pathohistological analysis showed fibro-cellular stroma with scattered islands of osteoid tissue varying in size (both mature lamellar bone and immature bone). Calcification of the bone tissue showed peripheral osteoblastic margins in some places. Some proliferations of the endothelial and inflammatory cells were present in the tissue. Overall, the pathohistological characteristics indicated POF [Gulati et al. reported a case of a female, 56-year-old patient with a mass of hard consistency upon palpation. The dimensions were 3.5 cm × 4 cm × 3 cm. The mass was located near teeth 13–23 and the teeth were completely periodontally compromised (stage III). A mass had a wide base and was erythematous and painless. The panoramic radiograph showed a clean radiographic status. 940-nm diode laser  .Interestingly, Sudhakar et al. show the case of a 55-year-old woman with a mass in the upper jaw in the second left incisor. An intra-oral periapical radiograph (IOPAR) and orthopantomography did not reveal any pathological changes except for generalized horizontal bone loss. Similar to the pathohistological finding in the patient shown in our case, the underlying connective tissue was highly cellular, with plump fibroblasts intermingled in a delicate fibrillar stroma associated with areas of woven tra-becullar bone and osteoids.Furthermore, due to the gender of the patient and the second decade predilection, the role of hormones has also been questioned as the predisposing factor of POF . It is important to point out that, in our case the patient, is a man in his seventies, with the appearance of POF in the area of the left lateral part of the mandible. In the literature that we have already mentioned, the occurrence of POF as well as other ossifying or cemento-ossifying fibromas is more common in females aged from 20 to 50 years. Predilection sites of neoplasms are most common in the front of the maxilla or in the area of the hard palate in the distal parts of the maxilla.Agarwal et al. Present a 68-year-old female patient troubled by a soft tissue growth on her left palate. The patient noticed a small nodule that grew to the present size in 4 months. The patient had no significant medical and personal history. Clinical intraoral examination discovered a shiny, rounded and elongated pink enlargement on the left side posterior of the palate in projection of the cementoenamel junction of 26. It extended from 23 to 27 anteroposteriorly and was 1 cm lateral to palatal midline to the occlusal surface of the left maxillary molars, buccolingually. Histological examination revealed connective tissue stroma with a rich fibroblastic nature and overlying epithelium comprising bony trabeculae with osteoblastic rimming of mature bone with a mostly lamellar structure, confirming the lesion as POF. On regular follow-up, the lesion healed without any complications [Katanec et al. published a similar case of symmetrical fibrous hyperplasia of the palate. In that case, a 47-year-old patient developed a bilateral mass in the hard palate, spreading to the junction of the hard and soft palate. A fibromatous mass appeared 3 years before the visit to the clinic. One year before the clinical examination, the nodule grew to form a voluminous fibromatous mass larger than 5 cm in diameter on both sides. The mass affected the area of the upper canines on both sides to the border with the soft palate. The formation was hard to palpation and connected at a wide base to the palatal artery. No signs of acute inflammation were present. Excision of the formation without interference with healthy tissue, followed by the removal of the affected periosteum and periodontal ligament, was the most suitable treatment. It is of the utmost importance to limit the irritating factors and possible trauma to the tissue .Dutra et al. concluded that the incidence of hyperplastic lesions in all oral pathogenesis is high. It is more common in the female population on the gingiva in the anterior part of alveolar ridge. Inflammatory fibrous hyperplasia is the most common lesion (72.09%), followed by oral pyogenic granuloma (11.79%), giant cell fibroma (7.30%) and peripheral ossifying fibroma (5.24%) .Borghesi et al. present the appearance of four benign formations in the same hemimandible, diagnosed by CBCT in a 50-year-old female patient. That was the first case in the literature showing peripheral osteoma (PO), compound odontoma (CO), focal cemento-osseous dysplasia (FocCOD), and cemento-ossifying fibroma (COF) together in the same patient mandible. This research, as well as our case, is of great importance for the case of differential diagnosis of neoplasms in the oral cavity and bones .In a retrospective survey study and literature review, Sangle et al. suggest that traumatic fibromas are the clinically most common lesions in oral cavity. As we have already stated, Sangle et al. concluded that irritating factors are important predisposing elements for the occurrence of fibromatous formations .We presented a case report of a 70-year-old male patient who had histologically confirmed POF in left mandible lingually. As elaborated in the discussion of this paper, this case is interesting because POF is more often found in female patients aged from 20 to 30 years. This presented case report is interesting as the patient is an adipose man in his seventies, who has two Toronto bridges placed on four implants in his upper and lower jaw. The occlusion of the bite surfaces of the bridges was not adequately aligned, which led to irritation and was a possible predisposing factor for the development of irritative fibromatosis, i.e., POF in this case.Overall blood count showed elevated parathyroid hormone levels. This fact may also be related to the formation of POF. After surgical excision of the formation, the operated area of the patient healed properly. There are no signs of recurrence at present, and the occlusion of the existing bridges is properly coordinated.


Here, we report a 41‐year‐old female with type II VUF after hysteromyomectomy.We report the diagnosis of VUF by imaging method, and provide a feasible treatment for this complication after pelvic surgery. As a urogenital fistula, vesicouterine fistula (VUF) is more common in female patients after pelvic surgery. In this study, we report a 41‐year‐old woman with type II VUF after hysteromyomectomy. Jówik2A 41‐year‐old female patient came to the hospital for treatment because of “hematuria during menstruation with vaginal leakage for 2 years.” The patient underwent a “hysteromyomectomy” two years ago. After the operation, it was found that there was obvious abdominal pain and gross hematuria during menstruation and occasional vaginal leakage. She tried to relieve her symptoms by placing an IUD, but the symptoms of abdominal pain and periodic hematuria did not improve. Routine urine examination in our hospital showed the following: red blood cells (++++); ultrasound showed that the continuity of the anterior wall of the cervix was interrupted at the level of the internal cervix, the defect was 0.55 cm wide, and the cervical canal communicated with the bladder Figure ; enhance3VUF accounts for only 1%–4% of all urogenital fistulas, and the increase in cesarean section is the main reason.4In this study, we present a clinical case report, type Ⅱ VUF, and it is recommended that MRI examination +cystoscopy + contrast medium bladder perfusion test be used for qualitative and localized diagnosis before operation. The treatment plan for urogenital fistula is to take active intervention measures. When fistula is found after delivery or operation, it is recommended to carry out conservative treatment for at least 4–8 weeks through bladder intubation and promote fistula healing through oral gonadotropin drugs. If fistula is too large (>2 cm), surgical repair should be carried out as soon as possible to prevent the formation of old fistula.There are no conflicts of interest in the paper.ZP‐Y contributed to research conception and drafted the manuscript. CC‐W contributed to literature search and review. YY‐W participated in data collection. ZP‐Y and CC‐W should be considered joint first authors. All authors have approved the submitted manuscript and accepted all revisions.Written informed consent has been obtained from the patient.


We describe two cases of diaphragmatic endometriosis treated using the robotic assisted laparoscopic approach, in which an incidental tension pneumothorax occurred during the initial inspection and assessment of diaphragmatic lesions. We demonstrate the importance of early diagnosis of this complication and report successful resolution using the thoracic drainage technique. In case one, after the pneumoperitoneum was installed, during the cavity assessment and inspection, small endometriotic lesions were observed in the tendon portion of the diaphragmatic surface. We observed a sudden increase in maximum airway pressures and a reduction in tidal volume, associated with arterial hypotension and hemodynamic instability and bulging of the diaphragm, which led to the diagnosis of a tension pneumothorax. In case two, diaphragmatic endometriotic lesions were also observed after hepatic mobilisation and following visualisation of the endometriotic lesions, an abrupt decrease in the capnography values was observed, consistent with hypertensive pneumothorax. In both cases, even after deflation of the abdominal cavity, hemodynamic instability persisted. We treated both cases with thoracic drainage, which immediately normalised respiratory parameters and resulted in hemodynamic stabilisation, and the surgical procedures were continued. During laparoscopic procedures for the treatment of diaphragmatic endometriosis, the endometriotic lesions can behave as communication hole in the tendon portion of the diaphragmatic surface and the changes in ventilatory patterns and haemodynamic instability should alert the medical team to the development of an incidental tension pneumothorax. The early identification of this complication in both cases allowed rapid intervention for chest drainage and allowed the surgical procedure to continue. Endometriosis is a disease characterised by a chronic oestrogen dependent inflammatory process that mainly affects the pelvic organs. Multiple theories have been postulated about its aetiology. One of the most accepted theories describes how the detached endometrial cells travel retrograde to the pelvic organs . In the A 38 year old nulliparous patient with a BMI of 20 and ASA I classification was diagnosed with infiltrating pelvic endometriosis . She gave a history of chronic pelvic pain, infertility, and right sub- scapular pain that worsened in the menstrual period. Previous surgical history included a laparoscopic rectosigmoidectomy, bilateral ureterolysis, left excision of ovarian endometrioma, left nerve decompression and bladder nodule shaving performed in another hospital 1 year previous. Following the initial laparoscopic procedure she developed sepsis and a right pleural effusion which was drained. She remained hospitalised for 23 days with persistent symptoms very similar to those that led her to initially seek medical attention. She underwent further surgery for the treatment of infiltrative pelvic endometriosis six months later with shaving of a rectal nodule, left ovarian implant vaporisation and excision of posterior cul-de-sac endometriosis implants. During laparoscopic assessment of the abdominal cavity, lesions of endometriosis were found on the diaphragmatic surface, but were not treated due to the potential higher infection risk due to intestinal manipulation and prolonged surgical time. After 6 months of medical treatment with suppression of menstruation with GnRH analogues (goserelin 3.6 mg monthly subcutaneously) and add back hormone therapy  for six months, upper abdominal magnetic resonance imaging (MRI) was performed. This did not detect any endometriotic lesions in the diaphragm and she was booked for second look surgery.2 insufflator was switched off and a chest drain was placed in the fourth right intercostal space, mid-axillary line. After drainage, there was an immediate normalisation of the maximum airway pressures and hemodynamic stabilisation. With effective drainage, the abdominal cavity was insufflated with 15 mmhg and surgical treatment of the respective lesions was continued, assisted by robotic laparoscopy. To treat diaphragmatic lesions, monopulmonary ventilation was installed and video-assisted thoracoscopy was performed to aid in resection of the lesions, delimitation of the surgical margin, and identification of important anatomical structures  for proper management and reconstruction of the diaphragmatic defect. Using a laparoscopic approach, the right falciform and triangular ligaments were dissected, with resection of a multi-fenestrated endometriotic lesion in the central tendon of the right diaphragm measuring approximately 8 cm, followed by reconstruction using robotic assisted laparoscopic suture. The surgery time was 200 minutes.In the operating room, selective tracheal intubation was performed with a 35F double-lumen tube on the right, with no evidence of complications in the respiratory tract. The correct positioning of the double light tube was confirmed by pulmonary auscultation, showing effective selective ventilation. After proper positioning (standard position for pelvic and thoracic access) pneumoperitoneum was created with an initial intracavitary pressure of 15 mmHg. The cavity assessment was performed with the patient in the prone position, and after slight hepatic mobilisation, endometriotic implants were visualised on the right diaphragmatic surface , comprom2) was noted, concomitantly with arterial hypotension, maintaining adequate saturation of oxygen. With direct laparoscopic vision, multiple holes were found in the diaphragm in the endometriotic lesions, which acted as communicating lesions during hepatic mobilisation. As in the previous case immediate desufflation of the abdominal cavity and treatment of the tension pneumothorax were performed  (A 45-year-old nulliparous patient with a BMI of 25.6 and ASA grade II presented to the department with a diagnosis of deep endometriosis (ASMR IV ENZIAN :A3 B2 C3 FO FU FI). She was booked for a surgical treatment of pelvic and diaphragmatic endometriosis following suppression of menstruation with GnRH analogues. The planned surgery included a robotic laparoscopic resection of diaphragmatic lesions assisted by video- thoracoscopy. While pelvic inspection was being performed, an abrupt change in capnography (COhe lung) . After dThe incidence of diaphragmatic endometriosis is uncommon and difficult to diagnose . In mostDiaphragmatic fenestrations in endometriotic tissue are described in the literature as a mechanism of pneumothorax formation, being responsible for 7.3-36% of cases of spontaneous catamenial pneumothorax . Althoug2 supply to the right heart chambers. Low cardiac output (shock) reduces perfusion of the alveolar segments, which therefore do not participate in gas exchange. Furthermore, collapse of the lung areas due to pneumothorax can lead to an abrupt decrease in EtC02 values on capnography, which is a warning sign found in diaphragmatic endometriotic resections. In the second reported case, no significant increase in airway pressure was detected, but the reduction in EtC02 values served as a parameter for diagnostic suspicion. In a case report of pneumothorax during anaesthesia for puncture of an ultrasound-guided breast tumor, reduction of EtCO2 was also one of the first indicative signs of pneumothorax . Such conditions should be considered, since prompt diagnosis and treatment are necessary to avoid serious repercussions and even death. In both cases, thoracic drainage was chosen, which caused immediate normalisation of respiratory parameters and hemodynamic stabilisation, allowing surgery to continue.In both cases presented, the simple movement of the liver to visualise the lesions, associated with the maintenance pressure of the pneumoperitoneum, was enough to open communication channels in the endometrial tissue and the formation of tension pneumothorax, even without surgical manipulation of the endometriosis lesions. Diaphragmatic endometriotic tissue remains a possible route of communication with the chest, and with increasing pressures in the abdominal cavity, suspected pneumothorax should be considered with clinical signs suggest this. Capnography indirectly reflects the state of pulmonary circulation and COIntraoperative pneumothorax is a complication that can occur in patients with pelvic endometriosis treated with any kind of minimally invasive approach as diaphragmatic endometriosis is often under-diagnosed by imaging tests. The anesthesiologist and the surgeon should be aware that such a complication is a possibility for these patients and should be prepared to diagnose and treat this complication immediately.


Intestinal Malrotation is an uncommon entity in the adult population; more so in monozygotic twins, where concordance is expected. In literature, discordant intestinal Malrotation has only been discovered when one twin became symptomatic, and the other was screened. To the best of our knowledge, this is the first documented case of discordant adult type intestinal Malrotation in otherwise asymptomatic monozygotic twins discovered incidentally during Laparoscopic Roux-en-Y Gastric bypass (LRYGB).Twins A and B met the NIH criteria for bariatric surgery, neither having symptoms of acute or chronic volvulus or history of intraabdominal surgery. Twin A had a LRYGB performed by a trained bariatric surgeon, noting no anatomic anomalies. 5 months later, Twin B had LRYGB and intestinal Malrotation was diagnosed incidentally.Diagnosis of Intestinal Malrotation is rare in adults, usually discovered after becoming symptomatic or during abdominal imaging for another indication. Two cases of discordant intestinal Malrotation in monozygotic twins have been documented, both discovered when one twin became symptomatic due to acute volvulus, suggesting epigenetic phenomena. When discovered incidentally during surgery, patients can safely undergo their intended procedure, but literature suggests prophylactic division of Ladd's bands, while appendectomy is left to the discretion of the surgeon.Intestinal Malrotation appears to be associated with epigenetic phenomena and if discovered incidentally during surgery, the proposed procedure can be carried out by an experienced surgeon, in addition to division of Ladd's bands and appendectomy. •Discovering Intestinal malrotation is rare in adults.•Concordant intestinal malrotation is expected in monozygotic twins.•Incidental finding of discordant intestinal malrotation in monozygotic twins.•Discovered during laparoscopic Roux-en-Y Gastric Bypass on second twin.•We report the first documented case. The first case involved an infant, and the second involved a 33-year-old female This case report has been reported in line with the SCARE Criteria. [6]2A pair of 30-year-old, homozygous twins, A and B, , both gave a history of obesity, with obstructive sleep apnoea and gastroesophageal reflux disease and met the NIH criteria for bariatric surgery after comprehensive multi-disciplinary team evaluation Twin A had a standard LRYGB performed by a board-certified bariatric surgeon who noted no anatomic anomalies . The proFive months later, Twin B underwent LRYGB using a standard 6-port technique by the same surgeon. The technique used creates the gastric pouch first, followed by the jejuno-jejunostomy, then the ante-colic gastro-jejunostomy.In preparation for the Roux and Y limbs, the small bowel was examined in search of the DJ flexure. There was difficulty whereby the DJ flexure was not easily identifiable when elevating the transverse colon . The appConsidering the intraoperative findings, the patient's history was retaken, but there were no findings suggestive of malrotation, with unremarkable family, genetic and psychosocial histories.At 12 months post procedures, both patients saw the desired weight loss , Fig. 4 3After an extensive literature search, we present the first documented case of incidentally discovered, discordant, isolated intestinal Malrotation in asymptomatic adult twins during laparoscopic surgery. Malrotation in adults accounts for 0.2–0.5% of cases, of which 15% present with midgut volvulus o and 90o, respectively. In stage two, the bowel returns into the abdominal cavity (week 10). The Duodeno-Jejunal and Caeco-colic loops continue to rotate, both completing 270o total rotation. In Stage three (week 11 onward), the cecum descends to the right lower quadrant, and there is fixation of the mesenteries. Abnormalities of rotation are classified as nonrotation (Arrest in Stage One), incomplete rotation , or incomplete fixation (Arrest in Stage Three). All carry the risk of midgut volvulus Under normal circumstances, the bowel rotates around the superior mesenteric artery in stages. Stage one (5–10 weeks gestation) involves physiological herniation of the bowel and anticlockwise rotation of the DJ loop and the CC loop 180The aetiology of intestinal Malrotation is incompletely understood, but authors have suggested a genetic component. A review by Martin et al. suggested the involvement of heterozygous mutations in the forkhead transcription factor FOXF1 and mutations in genes controlling L-R patterning; however, these are associated with other specific syndromic changes Intestinal Malrotation in monozygotic twins is rare. Kikuchi et al. first documented a case of concordant Malrotation in 1977, in new-born twins presenting with bilious vomiting Two cases of discordant intestinal Malrotation in monozygotic twins have been reported. Smith et al. (2005) Levin et al. suggest screening of the asymptomatic twin when Malrotation is discovered in the other. The potential loss of bowel offsets the additional cost, and potential risk of radiation from screening should a midgut volvulus occur Typically, patients diagnosed during adulthood are asymptomatic or have a history of chronic abdominal pain and vomiting . Controversy exists concerning the proper management of asymptomatic patients in whom Malrotation is discovered incidentally during imaging/surgery for another indication, as the risks of corrective procedures in this population are not known Our patient was asymptomatic and 30 years old, and we opted to perform LRYGB, division of Ladd bands and an appendectomy.4Malrotation, though once thought to result from specific genetic abnormalities and autosomal inheritance patterns, appears to be associated with epigenetic phenomena. Evidence supports screening an asymptomatic twin if Malrotation is discovered in the other. If asymptomatic malrotation is discovered before the age of 20, a prophylactic Ladd's Procedure is warranted. When this is discovered during bariatric surgery, performing the proposed procedure is generally safe but should be performed by an experienced surgeon, should difficulty be encountered. Ladd Bands should be divided where possible, and an appendectomy may be performed at the surgeon's discretion.None.This report is not a research study.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.Dr. Ryan Nicholas.Not commissioned, externally peer-reviewed.Dr Ryan Nicholas: Conceptualization, Writing- original draft, review and editingDr Sidiyq Mohammed: Writing- Review and editingDr Nigel Bascombe: VisualizationDr Dilip Dan: Surgeon who performed operations and follow-up, Supervision, Project Administration, Writing- Review and EditingThe authors have no conflicts of interest to declare.


Cases of acute scrotum presenting to the emergency department are usually due to testicular torsion, complicated hernia, or epididymo-orchitis. Thrombosis of the spermatic vein is an uncommon entity and most of the cases reported to date are of unilateral involvement. Here, we present an extremely rare case of spontaneous bilateral spermatic vein thrombosis, which was diagnosed by a Doppler ultrasound of the testes. This article highlights its clinical presentation, radiological imaging, etiology, and management. Spermatic vein thrombosis is an uncommon event and needs thorough evaluation for differential diagnosis. The acute scrotum is the presenting complaint in most cases . IsolateA 38-year-old gentleman came to the emergency department with complaints of sudden onset of bilateral scrotal pain and lower abdominal pain. There was no history of vomiting, dysuria, obvious trigger, or similar illness in the past. He had transurethral resection of the prostate (TURS) and aspiration of prostatic abscess six years ago. The patient was a smoker and was working as a laborer carrying heavy objects. On examination, his vitals were stable. Local examinations showed no scrotal swelling. Mild erythema was present over the scrotum. Testes were firm, non-tender, and without masses or lesions. Other systemic examinations were within the normal limit. Basic blood investigations and coagulation profiles were normal.A Doppler ultrasound of the testes was done from the emergency department to rule out testicular torsion, which showed bilateral testes with normal echogenicity and vascularity Figure . Long seComputed tomography of the abdomen and pelvis showed no mass lesion. CT angiogram of the abdomen was done to assess the extent of spermatic vein thrombosis and to know if there is any other thrombus, which revealed patent major abdominal vessels with no evidence of filling defect. The patient was started on rivaroxaban 15 mg, twice daily for three weeks, followed by once a day for three to six months. The patient became symptomatically better after five days in the hospital and was discharged home with rivaroxaban tablets and anticoagulation clinic follow-up.There are 42 reports of various thromboses of the pampiniform plexus, spermatic vein, and/or testicular vein found in the literature. Only two cases were found to be involving bilateral spermatic veins thrombosis and the most common site of occurrence was on the left side in other cases. Bilateral pampiniform plexus thrombosis reported by Kamel et al. was caused by protein C deficiency, whereas Bakshi described an idiopathic case mimicking incarcerated hernia ,2. The lAll venous thromboses in some form or the other have been attributed to one of Virchow’s triads, which are endothelial damage, blood flow stasis, and hypercoagulability. Our patient had been working as a laborer carrying heavy objects for more than eight hours per day, which increased intra-abdominal pressure that might have reduced the spermatic blood flow causing stasis and thrombosis. Other factors were excluded as coagulation profiles were normal and there was no recent surgical history or trauma.The low blood flow in the left testicular vein as it joins the left renal vein is the cause of left dominance of spermatic vein thrombosis, and any cause of decreased flow in the testicular vein such as genitourinary malignancy or conditions like nutcracker syndrome might lead to spermatic cord thrombosis . IsolateThe progression of spermatic vein thrombosis is variable; it can go away on its own, or it can be worsened by pulmonary embolism . ConservSpontaneous bilateral spermatic venous thrombosis is an uncommon entity presenting as the acute scrotum. Therefore, a high threshold of suspicion is required as not to miss other causes of acute scrotal pain like torsion and incarcerated hernia, which will need surgical intervention. Doppler ultrasound is the first choice of investigation since it can also be used to assess other differentials. In patients with acute scrotal pain, it helps clinicians distinguish between those who need surgery and those who can benefit from conservative treatment alone. Conservative management is the initial treatment for bilateral spermatic vein thrombosis, which includes pain killers and anticoagulants.


Background: The Caldwell-Luc (CL) procedure, an outdated operative procedure that is used to treat inflammatory sinus diseases, is rarely performed presently. However, physicians may encounter patients with a history of CL surgery who develop considerable postoperative changes that may lead to diagnostic confusion in imaging evaluation; increase the difficulty of future surgery, such as sinonasal surgery; and increase the incidence of future intraoperative complications. Case summary: A 67-year-old man with a surgical history of chronic sinusitis reported epiphora of the left eye for five years. Balloon dacryocystoplasty was attempted but failed. Endo-DCR (Endoscopic dacryocystorhinostomy) was indicated; however, preoperative CT (computed tomography) imaging and nasal endoscopic examination showed sinonasal anomalies and the loss of internal landmarks for localizing the lacrimal sac. Preoperative CT results indicated previous CL surgery. Endo-DCR was performed with the aid of nasal forceps and a 20-gauge vitreoretinal fiberoptic endoilluminator. A six-month follow-up revealed the complete resolution of symptoms and no signs of recurrence. Conclusions: Epiphora might be a delayed complication of the CL procedure. Before performing endo-DCR, ophthalmologists should be familiar with the sinonasal anatomy and carefully assess preoperative imaging to identify anatomical variations. Nasal forceps and transcanalicular illumination can assist in determining the precise location of the lacrimal sac during endo-DCR. Endoscopic dacryocystorhinostomy (endo-DCR) is indicated for the restoration of tear drainage when the lacrimal pathway obstruction is at or below the level of the lacrimal sac. Compared with external DCR, endo-DCR has comparable long-term outcome and the advantages of avoiding external skin incision, preserving the pumping function of the orbicularis muscle, and permitting access for the treatment of concomitant sinonasal problems simultaneously ,2,3,4,5.The Caldwell-Luc (CL) approach, described originally by George Caldwell and Henri Luc more than a century ago, is the main procedure for approaching the maxillary sinus and nearby structures before the advocates of functional endoscopic sinus surgery (FESS) ,8. The oHerein, we present a challenging case of nasolacrimal duct obstruction (NLDO) with a post-CL-procedure and the associated distorted intranasal anatomy receiving endo-DCR, and the evaluation of the preoperative assessment, computed tomography (CT) findings, and surgical techniques.A 67-year-old man presented with hypertension, diabetes mellitus, and a surgical history of left chronic sinusitis more than 50 years ago. Three years ago, he presented to our outpatient clinic because of tearing of his left eye for two years. According to medical records, a diagnostic probing test revealed a hard stop. Balloon dacryocystoplasty was performed but failed, and no further intervention was conducted due to loss to follow-up. This time, he returned with worsening epiphora and a new-onset yellowish mucopurulent discharge in the same eye.An anterior segment assessment revealed a clear cornea with a high tear meniscus height in his left eye. There was no swelling of the medial canthus or abnormality of the eyelids, including lid laxity, ectropion, entropion, punctum eversion, and blepharitis. Lacrimal irrigation was performed; however, mucopurulent reflux through the upper punctum was noted. A dye disappearance test demonstrated delayed fluorescein clearance. Further diagnostic probing showed a hard stop, indicating the presence of a patent common canaliculus with an obstructed lacrimal sac or nasolacrimal duct. After discussing with the patient, he agreed to undergo endo-DCR surgery.As the patient had a surgical history of chronic sinusitis, a preoperative non-contrast computed tomography (CT) scanning (2-mm section) was performed to analyze the anatomical alterations. The imaging findings showed canine fossa bony defect, bony opening of the anterior and medial walls of the maxillary sinus, sclerosis, destruction and wall thickening of the maxillary sinus, and a collapse of the maxillary sinus cavity with distinct sinus volume reduction and increased orbital volume on the left. An expanded left lacrimal sac and a dilated nasolacrimal duct due to obliterated terminus were noted. Surgical landmarks, such as the maxillary line, axilla, uncinate process, and agger nasi, that are used to guide endo-DCR were absent. In addition, a minimally preserved MT was observed . A largeThe preoperative nasal endoscopic examination that was performed by a rhinologist showed polypoidal changes in the nasal septum, consolidation of the maxillary sinus, and enlarged space of the left nasal cavity due to the decreased size of the middle concha. No sinusitis was observed .®; Cross Instruments Inc., Edmonton, Canada) through both sides of the left nasal ala, with the outer tip of the forceps aligned with the medial canthus where the lacrimal sac was located. Subsequently, we lightly clipped together the inner tip, which then pointed to the corresponding intranasal position of the lacrimal sac  were inserted through the superior and inferior puncta into the nasal cavity. The whole procedure was performed smoothly without complications, and anatomical and functional success was achieved. A six-month follow-up revealed complete remission of the epiphora. The patient was quite satisfied with the surgical outcome as he was free from tearing without any postoperative external deformity.The patient’s MT and axilla of the MT, which are the internal surface landmarks of the lacrimal sac, were absent. Thus, during the endo-DCR, we inserted nasal forceps (Jansen Bayonet Nasal/Ear Forcepsimal sac A,B. Afteimal sac . The lacThe CL procedure, also known as radical antrostomy, which was pioneered by George Caldwell and Henri Luc more than 100 years ago, is outdated ,8. In thThe maxillary sinus is the biggest paranasal sinus with a medial wall lying adjacent to nasal cavity and is the major site where the CL procedure is performed . The chaThe CL procedure, although generally safe, is not without risk of complications. Recurrent sinusitis, facial and gum numbness/pain, oroantral fistula, and dental problems have been reported as complications of CL ,15,16. EEarlier literature suggested that external DCR is the procedure of choice for persistent NLDO post-CL procedure ,19,20. IWe reported a case of NLDO with characteristic CT findings after the CL procedure. The preoperative evaluation with CT is beneficial for identifying the anatomic variants, making appropriate assessments, and preparing precise surgical plans, and, thus, reducing possible complication rates. The combined use of nasal forceps and lacrimal illuminator allows a quick and easy way to localize the lacrimal sac during endo-DCR in cases of sinonasal anomalies. Surgical success and complete resolution of epiphora symptoms were achieved.


Hidradenoma papilliferum is a rare benign neoplasm arising from apocrine glands. It occurs commonly on the anogenital region of middle-aged women. It usually presents as a slow growing, solitary asymptomatic, skin colored or red nodule less than 1 cm in diameter.The case is a 38-year-old, white woman who presented with a painful nodule occurring within a month in the himenal region of the posterior vaginal introitus. The nodule was excisied and the histology revealed a hidradenoma papilliferum. The diagnosis and treatment of hidradenoma papilliferum is possible with surgical removal and histopathological evaluation of nodules.When an adult woman presents with a noduler lesion in the anogenital area, sexually transmitted diseases and other benign and malignant vulvar lesions, as well as malignant transformation is very rare but,should be kept in mind; however because it has been reported and long-term clinical follow-up is suggested Hidradenoma papilliferum is a rare, benign apocrine tumor that occurs almost exclusively in the anogenital region of midle-aged women; they are generally solid, asymptomatic, well-confined, skin colored or red, with a noduler appearance ranging in size from 0.5 cm to 1 cm . These nDyspareunia is a symptom of a variety of disease states which can have both organic and psychological dysfunction components. There are many ways of classifying dyspareunia; based on cause, onset, frequency or location. dyspareunia is found in etiological factors in vulva lesions.Here we reported hidradenoma papilliferum of the Hymen. When the literature was investigated, we found that this case is the first case of hydradenoma papilliferum in the hymenal region; therefore we aimed to discuss and present our case in the light of the literature.A 38 years old white woman with gravida 0 and no additional disease applied to our clinic for vulvar pain that has been going on for about 1 month. The patient did not have any additional disease and did not have any medication. She stated that she had pain in particular during sexual intercourse and her pain persisted for a few more hours after sexual intercourse. She also stated that she had a hard structure at the entrance of the vagina. In the gynecological examination of the case; hymen at 6 o'clock, nodular, approximately 1 × 2 cm, with regular borders, painful solid lesion on palpation  . Local eWhen an adult woman presents with a noduler lesion in the anogenital area, sexually transmitted diseases and other benign and malignant vulvar lesions, as well as Hidradenoma papilliferum should be kept in mind. Patient history and clinical findings are not specific for hidradenoma papilliferum and surgical removal of the lesion and its histopathological evaluation are required to make a definitive diagnosis.