Inguinal bladder hernias are rare incidents accounting for 1–3% of all inguinal hernia. Most of those cases are discovered intraoperatively accounting for the high incidence of bladder injuries during the repair. Symptoms can be variable depending on the size of the herniated bladder.We present here a 70 years old obese male patient, with history of CVA & on regular Aspirin ingestion. He presented to the ER as a case of intestinal obstruction due to suspicion of strangulated inguinal hernia based on an unclear Ultrasound picture. He underwent an emergency exploration of the hernia where the balloon of a Foley’s catheter was found inside a diverticulum of the urinary bladder, herniated through an Ogilvie hernia.Ogilvie hernia is a rare incident that happens mostly in elderly males. The herniated part contains prevesical fat, bladder andor loops of intestines. Diagnosis must be made preoperatively through CT scan or Cystourethrogram. First line management involves surgical reduction of the bladder and hernia repair. Resection of the herniated part is generally limited to necrotic tissues.Surgeons must have high index of suspicion for elderly obese males with inguinal hernias and urinary or intestinal obstruction symptoms. Careful preoperative planning must be made to avoid repair associated bladder injuries. It is gOgilvie’s inguinal hernia, which we present here, is caused by a small defect in the medial part of conjoint tendon and above the pubic tubercle. It usually contains prevesical fat and part of urinary bladder ,5. It caDespite the advancement in diagnostic technologies and since most of the cases are asymptomatic, it is still a challenge to diagnose inguinal bladder hernia where <7% is diagnosed preoperatively and many of the cases are identified incidentally .Patients with complete bladder herniation will present with double voiding which means the patient should compress his scrotum to fully evacuate the bladder . InguinaIn this prospective, single Centre, Case report study, we present a case of 74-year-old male who presented with sliding direct Ogilvie inguinal hernia and upon surgical exploration a Foley’s catheter was found in the herniated part of the bladder. The case was managed in Al Bashir teaching hospital in Amman, Jordan in 2010 and was followed up in the same hospital. This paper was written according to SCARE criteria for case reports .2A 74-year-old obese male was presented to ED (Emergency Department) with abdominal pain, distension, vomiting and diarrhea for 5 days. His past medical history is significant for hypertension and CVA (Cerebrovascular accident) 3 years before admission. He had a Colonic polyp removed 5 years ago. In addition, He had a history of gallstones removed 10 years ago. He was on Atenolol. Captopril and, Aspirin. On examination, the patient had generalized weakness of the left side of his body due to previous CVA.There was a tender irreducible swelling in the right inguinal region, covered by normal skin. Since the patient had a history of stroke years ago, it was not possible to ask him about duration of the swelling, pain at the site, or to cough to evaluate the swelling more. The vital signs were within normal. Bowel sounds were sluggish. Digital rectal examination showed nothing of significance.3/mm3, Hemoglobin: 12.4 g/dL, Platelets: 312 × 103/mm3.Lab results showed Blood Urea: 11.8 mmol/L, Serum Creatinine: 116.96 μmol/L, Serum Potassium: 5.5 mmol/L, Blood Sugar: 10.3 mmol/L, WBC Count: 14.2 × 10On Abdominal ultrasound, the gallbladder wasn’t visualized, but a cystic like lesion in the right inguinal region mostly representing a bowel loop was seen. The tentative diagnosis for this case was strangulated right inguinal hernia causing intestinal obstruction. Consent was taken from the patient’s next of kin and the right inguinal region was explored. A sliding direct Ogilvie inguinal hernia was discovered. There was a well circumscribed soft mass of a narrow neck, protruding from the posterior wall of the hernia that looks like a foreign body . On expl3Studies have shown that Bladder hernia is uncommon to happen, constituting only 1–3% of all inguinal hernias . The maiOgilvie hernia is a much rarer type of hernias which usually occurs in elderly males, as in our case. It’s characterized by a narrow short neck. Prevesical fat, portion of the bladder andor intestines usually are found herniating through a defect in the medial part of the conjoint tendon, just above the pubic tubercle. No epidemiological data about this type is reported in literature ,5.The presence of urinary symptoms depends on the size of the herniated part of the bladder.  Most of Less than 7% of all Foley’s catheter cases are diagnosed preoperatively, 16% are diagnosed postoperatively due to the complications, while 77% of the cases, including our case, are diagnosed intraoperatively. The importance of diagnosing bladder hernias before the operation lies in the high risk of damaging the bladder (12%) during the repair, which is significantly reduced if preoperative diagnosis was made [Gold standard imaging modality for diagnosis of Bladder hernias is Cystourethrogram. This modality is often used to assess the cases of outlet obstruction, which might or might not be seen in cases of bladder hernias. ,11. UltrBladder hernias can be managed conservatively by catheterization and decompression of the bladder  or by suSurgery involves resecting or reducing the herniated part of the bladder. Reduction is generally preferred to preserve the bladder volume and to prevent the need to incise and suture the bladder increasing the risk of infection. Resection is performed only in cases of necrotic herniated parts of bladder, small hernia neck (<0.5 cm) and bladder tumor ,11. In o4Inguinal Bladder Hernias are rare incidents commonly missed preoperatively. The symptoms can be variable depending on the size of the herniated part of the bladder. Surgeons must have high index of suspicion for elderly obese males with inguinal hernias and urinary or intestinal obstruction symptoms. Diagnosis must be made through imaging modalities and careful preoperative planning must be carried out to prevent bladder injuries.Author and Co-authors have nothing to disclose.This study was not funded by any sponsor.This study was exempted from ethical approval in Al Bashir teaching Hospital Amman. Jordan.A written informed consent was obtained from the patient’s next of kin. Copy of the written consent is available for review by the Editor-in-Chief of this journal on request.Study design: Amer Hashim Al Ani.Acquisition of data: Amer Hashim Al Ani.Analysis and interpretations: Amer Hashim Al Ani, Mohammad Bakri Hammami, Obaidah Adi.Manuscript draft: Mohammad Bakri Hammami, Obaidah Adi.Revision: Amer Hashim Al Ani.All authors have approved the final article.Registration Date : 09:09 March 11, 2018.Research Registry UIN: researchregistry3807.Primary Investigator and Sponsor Public and Scientific : Amer Al Ani.Public and scientific title of research : Foreign bodies in inguinal canal canal.Dr. Amer Hashim Al Ani.


Mesenteric cystic lymphangioma (MCL) is a benign malformation of the lymphatic system. MCL seldomly occurs with a very rare incidence of around 1: 250,000. This neoplasm is more common in pediatric patients. Symptoms can appear with various clinical features ranging from asymptomatic abdominal mass to acute abdomen.Here we report four pediatric patients with MCL: two males presented with bilious vomiting and ileus, one female with abdominal pain, and one female with asymptomatic abdominal mass. All patients underwent explorative laparotomy and mass excision was done and/or marsupialization. All patients were discharged at the third – fourth postoperative day uneventfully.Although benign, lymphangioma can cause other symptoms such as bleeding, torsion, or lymphangioma rupture. Therefore, MCL should be considered as one of the differential diagnoses in acute abdominal and abdominal mass cases in children.In conclusion, although sometimes asymptomatic and found incidentally, the MCL should be resected because it has the potential to grow and invade vital organs and cause life-threatening complications. This tumor is very rare with an incidence of 1: 250,000 and is commonly found in children, of which 60% appear at birth, and 40% by one year of age. MCL represent 5–6% of benign tumors in children [The predilection of tumors is in the head and neck (70%), axillary 20%), and internal organs (10%) . Diagnos0%, and iThe main therapy for MCL is mass resection with an open explorative laparotomy or laparoscopy approach. In some cases, MCL that is tightly attached to the intestinal wall might need an intestinal resection ,4. In th22.13 that pressed against the jejunum (Type 2 MCL)  A. The tue 2 MCL) B. Postop2.2A 4-year-old male was referred to our hospital for having an ileus. The patient’s complaints were bilious vomiting accompanied by bloating and abdominal pain for four days. The patient had a history of intermittent abdominal pain in the last one year before admission. On physical examination, abdominal distension was found. Abdominal X-ray was performed and showed small bowel obstruction A, wherea2.3A 3-year-old girl was referred to our hospital because of intra-abdominal tumors. Patient complained of a mass becoming bigger in the lower abdomen since the last three months accompanied by weight loss. On physical examination, abdominal distension was found with a palpable mass in the epigastric to the suprapubic area. Computerized tomography scans were performed and showed an oval tubular mass in the middle of the abdomen A. The pa2.43 that had entered the retroperitoneal area and attached in part to the transverse colon and descending colon (Type 4 MCL)  B. We per3Here, we reported four MCL cases with various clinical manifestations. Clinical features of MCL can vary, with the predominance of asymptomatic abdominal masses, but if complications arise, they can manifest as acute abdomen , depending on the size and location of the mass. Although benign, lymphangioma can cause other symptoms such as bleeding, torsion, or lymphangioma rupture. Therefore, MCL should be considered as one of the differential diagnoses in acute abdominal cases in children .MCL might be classified into four types as follows : Type 1)Some patients with lymphangiomas are reported to experience spontaneous regression. Drainage can be an alternative therapy for intraabdominal lymphangioma in patients at high risk but this procedure has a high risk of recurrence and the risk of perforation during drainage . Partial4Although asymptomatic and discovered incidentally, MCL needs to be resected because of the potential for growth and invasion of vital organs that can cause life-threatening complications. MCL total resection with clear microscopic boundary was the best approach if the facility provides the necessary supports.No funding was received for this study.This study has been approved by the Ethical Committee of Faculty of Medicine, Public Health and Nursing, Universitas Gadjah Mada/Dr. Sardjito Hospital (KE/FK/0569/EC/2019).Written informed consent was obtained from the patients’ parents for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on reasonable request.Gunadi conceived the study and writing the manuscript. Gibran Kashogi and Dedi Prasetya collected the data. Aditya Rifqi Fauzi, Eddy Daryanto, and Andi Dwihantoro critically revised the manuscript for intellectual content. Gunadi, Gibran Kashogi, Dedi Prasetya, Eddy Daryanto, and Andi Dwihantoro facilitated all project-related task.researchregistry5084.Gunadi.Not commissioned, externally peer-reviewed.Gibran Kashogi: Data curation. Dedi Prasetya: Data curation, Resources. Aditya Rifqi Fauzi: Writing - original draft. Eddy Daryanto: Data curation, Resources. Andi Dwihantoro: Data curation, Resources.Conceptualization, Investigation, Methodology, Supervision, Validation, Writing - review & editing. No potential conflict of interest relevant to this article was reported.


Dear Editor,We have for many years followed a patient whose long-term history illustrates how therapeutic decisions for benign lesions can affect and determine the entire life of a patient.In 1967, a 5-year-old girl presented with progressive headache, vomiting, and ataxia. Clinical examination disclosed papilledema. Ventriculography demonstrated supratentorial hydrocephalus and lack of contrast passage to the fourth ventricle indicating a posterior fossa tumor. She underwent resection of a large, partially cystic tumor. Postoperatively, there was a right-sided hypacusis and slight persistent ataxia.The histological examination showed astrocytoma WHO1. The resection was considered incomplete and she was given postoperative radiotherapy to the posterior fossa. Two posterior fossa fields were given small doses over 15 days up to 2750 r in each field.She recovered and after a couple of years, someone asked: “why did you give XRT?”.The answer was: “It was not much—only 2750 r x 2”Fourteen years later, in 1981, she once more presented with clinical symptoms and signs of intracranial hypertension. A CT scan revealed a large cystic expansion in the posterior fossa along with supratentorial hydrocephalus. After a second operation with opening of the cyst and subtotal tumor resection, the histology was once more astrocytoma grade WHO1. After this second procedure, she once more recovered, but experienced right-sided anacusis and over time progressive left-sided hypacusis as well.During the late 1990s, she deteriorated and MRI now disclosed large supratentorial meningioma formations. The tumor was resected along with the right side of the tentorium in 2000. Two years later, in 2002, she needed resection of a large meningioma in the sellar region and she was given hormonal substitution. During the following years, she lost her vision over time and underwent repeat tumor resections for aggressive malignant meningiomas in the years 2004 and 2006.The clinical condition deteriorated further and she was bedridden, deaf, and blind during the rest of life, until she passed away in 2016. Follow-up MRI in 2011 demonstrated further intracranial meningioma progression (Fig. Fifty years ago, postoperative radiotherapy in the treatment of posterior fossa medulloblastoma dramatically improved the 5-year survival rates from zero to more than 50% . In chilAlthough our policy has been repeat surgery when indicated instead of adjuvant radiotherapy, a few patients have been given such adjuvant postoperative radiotherapy up to the late 1980s [a kind of the worst possible result—a progressive disaster over nearly five decades.The present case is—in our opinion—


Rhabdomyosarcoma is uncommon in adults. Adult and maxillary rhabomyosarcoma with direct orbital extension has been rarely reported. To our knowledge, there is no reported case about adult patient with alveolar maxillary rhabdomyosarcoma and orbital extension survived 1 year with intact ocular function.A 21-year-old female presented with protrusion of the right eye and an obstructed nasal passage for the past month. Her symptoms were not relieved by oral antibiotic or irrigation. She was referred to our clinic. Computed tomography and magnetic resonance imaging showed a large homogenous well-enhanced mass with surrounding bony erosion and remodeling. The mass extended to the ipsilateral nasal cavity and orbit. Endoscopic biopsy of the nasal cavity confirmed alveolar rhabdomyosarcoma. The maxillary mass was excised using the Caldwell–Luc approach, and the orbital mass was excised using a transconjuctival and transcaruncular approach. A systemic work-up confirmed ipsilateral lymph node metastasis. The patient received 6 cycles of chemotherapy with vincristine, dactinomycin, and cyclophosphamide, as well as 5120 cGy radiotherapy. Her ocular function was intact 1 year after treatment, and magnetic resonance imaging showed complete regression of the tumor.Rhabdomyosarcoma, which is usually an aggressive malignancy, should be considered in the differential diagnosis of a rapidly growing orbital mass. Aggressive treatment, including surgery, chemotherapy, and radiation therapy, can increase local remission rates and improve the prognosis. The predilection sites of adult rhabdomyosarcoma are the extremities but the predilection sites in children with rhabdomyosarcoma are the head and neck area. Head and neck sites only account for 24% of adult rhabdomyosarcoma cases. Moreover, cases involving the orbit are extremely rare.Rhabdomyosarcoma is the most common primary orbital malignancy in children <15 years of age, accounting for approximately 5% of childhood malignancies. It is very difficult or impossible to surgically excise a mass within the maxillary area. When a tumor occurs in this region, it is usually more extensive locally or has metastasized distantly at the time of diagnosis. The 5-year survival rate is ≤8% in cases of head and neck rhabdomyosarcoma. Herein, we report the case of a 1-year survivor of adult alveolar rhabdomyosarcoma of the maxillary sinus with orbital extension.The prognosis of adult rhabdomyosarcoma is poor. More than 60% of adult patients have regional or distant metastasis at the initial diagnosis.Written informed consent was obtained from the patient for this case report. Ethical approval was obtained by the Institutional Review Board of Kyung Hee University Hospital at Gangdong (KHU-2010-07-39).2A 21-year-old female without specific medical history presented with a protruding right eye and an obstructed nasal passage of 1-month duration. The patient was diagnosed with sinusitis at another clinic and was prescribed oral antibiotics. Her symptoms were not relieved by the antibiotic treatment or nasal irrigation. She was referred to our clinic, and we performed a diagnostic work-up. Corrected visual acuity was 1.0 in both eyes. Hertel exophthalmometry revealed 3-mm proptosis (Fig. 3,6 Because of its rarity, no study has reported outcome statistics or an established therapeutic method. The bad prognosis for adults with orbital involvement is the difficulty excising the mass, the histopathological subtype, metastasis, and poor tolerance to intensive chemotherapy. Tumors in the paranasal sinus, nasopharynx, infratemporal palatine fossa, or orbit more often spread into surrounding tissues. These regions have a 35% incidence of meningeal recurrence. The alveolar subtype is more frequent in adults and the embryonic subtype is rare. The alveolar subtype has a higher metastatic rate than that of the embryonic or pleomorphic subtypes.,9,10 More than 60% of adult patients have regional or distant metastases at the initial diagnosis, and most rhabdomyosarcoma cases are classified as IRSG group 3. Therefore, the prognosis is poor because of impossible complete excision.Adult rhabdomyosarcoma rarely presents in adults. Orbital involvement is one of the most favorable factors in children, and the 5-year survival rate is >90%. A combination of chemotherapy and radiation therapy increases the local remission rate and decreases the metastatic rate. The conventional chemotherapeutic agents used are vincristine, dactinomycin, and cyclophosphamide. Darren et al suggested that 50 to 56 Gy was adequate for patients with completely resected disease and a negative resection margin. We also performed debulking surgery, conventional chemotherapy, and radiation therapy. There have been 7 recent cases similar to this case. Two cases died from systemic complications associated with the invasion. In other 2 reports, there was no mention of therapeutic response or progression because the case was in the middle of treatment.,14 The remaining 3 cases survived for >2 years.–17 Two cases were treated with chemotherapy. The other 1 case was treated with surgery and chemotherapy. Prognosis is worse in maxillary rhabdomyosarcoma than in other sinus rhabdomyosarcoma because extensive local disease is usually found at the time of diagnosis, making surgical excision difficult or impossible, and because local and distal metastasis is often found when treatment is initiated. There has been no recent report of a case of maxillary rhabdomyosarcoma with orbital extension in which a remission has been confirmed through imaging for 1 year. Although the duration of follow-up is short in our case, but the value of the report is considered sufficient.The treatment protocol for adults with rhabdomyosarcoma has not been established. Surgery is performed in most cases, and chemotherapy and radiotherapy are used as adjuncts following the pediatric treatment protocol. Surgery is used to debulk, reduce the mass effect on the optic nerve, and improve ocular motility.In conclusion, adult rhabdomyosarcoma is encountered very rarely in the orbit, where it is usually an aggressive malignancy with a poor prognosis. Rhabdomyosarcoma should be considered in the differential diagnosis of a rapidly growing orbital mass. Aggressive treatment, including surgery, chemotherapy, and radiation therapy can increase local remission rates and improve the prognosis.Conceptualization: Jin-Ho Joo.Data curation: Shin-Myeong Choi.Investigation: Shin-Myeong Choi.Supervision: In-Ki Park, Jae-Ho Shin.Writing – original draft: Jin-Ho Joo.Writing – review & editing: Ji Sang Han, Jae-Ho Shin.Jin-Ho Joo Orcid: 0000-0003-0846-6465.


Metastatic tumors of the penis are uncommon, and fewer than 500 cases have been reported since 1870. Most penile secondary tumors originate in organs of the genitourinary tract, followed by the gastrointestinal tract. Primary tumors of sarcoma origin are extremely rare. Herein, we present a teenager who had recurrent sarcoma of the right femur with penile metastasis.The 20-year-old male patient was diagnosed with sarcoma of the right femur when he was 16 days old. He was stable following combination chemotherapy with the VAC regimen  and debulking surgery. In January 2018, five months ago, he presented with right leg pain and swelling, and a recurrent tumor was found. Following excision of the tumor showed recurrent sarcoma. However, 2 months after the operation, right thigh swelling with tenderness occurred. A firm nodule on the glans of the penis was also noted. Penile metastasis was suspected and a biopsy was performed. The final pathology report disclosed pleomorphic sarcoma with penile metastasis. Symptoms including priapism and inguinal lymph node enlargement progressed rapidly within 2 weeks. He also complained of voiding difficulty with urine retention. The patient died 35 days after admission due to pneumonia with septic shock.Penile metasitasis largely occurs from organs in the pelvis. To the best of our knowledge, this is the first case of a teenager with a secondary penile tumor, metastasizing from sarcoma of the bone. It presented as a palpable mass, and then progressed into priapism. The patient had a dismal prognosis and the symptoms progressed faster than his physicians anticipated. Secondary penile tumors are a rare clinical entity, despite the rich vascularization and circulation between neighbouring organs. Fewer than 500 cases have been reported reviews since 1870 . The vasThe 20-year-old male patient was diagnosed with spindle cell sarcoma of the right femur in Aug. 1998, when he was 16 days old, at Mackay Memorial hospital (consent was obtained from his mother for the publication). Due to right thigh enlargement with a palpable mass, excisional biopsy were performed, and pathology disclosed a spindle cell sarcoma with hypercellularity  revealed a huge intraoseous cystic lesion in the right femoral shaft. Penile swelling was thought to be an balanitis at first. Urologists were then consulted for the penis lesion and 2 firm nodules about 1 to 2 cm in size were identified on the glans of the penis. No discharge, ulceration or necrosis was noted on the the overlying skin of the glans Fig. a,b,c. CoFour weeks after admission, the patient still had persistent priapism with gross hematuria, and poor appetite with progressive cachexia. The biopsy wound over the glans developed gangrene Fig. d. Fever The majority of secondary metastatic penile tumors origniate from adjacent organ systems in the pelvis, including the genitourinary organs or the rectum. Previous reviews reported that the most common metastatic organs were the prostate and bladder, followed by the colorectal system. Fewer than 10% of cases are from other organs, such as the lung, liver and bone .Nearly 500 cases of penile metastasis tumors have been reported, but only 3 previous cases have reported a primary tumor site with sarcoma origin. The first reported case was a 37-year-old male patient with penile metastasis from the primary site of chondrosarcoma in the jaw . The secTo the best of our knoweldge, the present study is the first case of sarcoma with multiple recurrences, where the first metastatic site was the penis. In addition, the present case is the first reported in a teenager. In a previous case series, the patients with penile metastasis were aged between 60 and 80 years  The retrIt is difficult to identify penile lesions except by an open biopsy. There is no classic presentation or symptom complex in penile metastasis. Our patient had unusual symptoms of priapism, urine retention and gross hematuria. Previous cases series, mostly presented with painless nodule lesions over the penis, followed by priapism and pain , 2, 9. HPenile metastasis is an indicator of a poor prognosis. The median survival time after the diagnosis of penile metastasis is 10 months (range 6–18 months) and the patient presenting with priapism and those with metastasis from non-urologic tumors have a significantly worse prognosis . Our 20-Secondary malignancy of the penis rarely occurs in young patients, and it mostly originates from the genitourinary organs. A literature review showed that the current case is the first case of metastatic penile tumor originateing from sarcoma of the femur in a teenager. He presented with a palpable mass and subsequently developed priapism and gross hematuria. Because the overall outcome is poor, most patients will need early palliative or supportive care.


A 10-week-old sexually intact female golden retriever was evaluated for two functional anal openings and a bipartite vulva. Examination revealed haired skin between two separate anatomically smaller anal openings. On rectal palpation, a soft tissue septum (5 cm) with a mucosal surface between the two anal openings was palpated. In addition, circumferential rectal musculature was not appreciated on the ventral aspect. Urogenital evaluation revealed duplication of the vestibule and vagina with a complete centrally located septum extending dorsoventrally. Computed tomography (CT) of the abdomen and pelvis, vaginocystourethrogram, and colonogram were performed. Complete bifurcation of the urinary bladder with duplication of the urethra, cervix, and vaginal canal was noted. Approximately 2 cm from the rectum, there was a similar bifurcation that converged the colon into two rectal portions and separate anal openings. The owner was counseled on the severity of congenital malformations and a high likelihood of aging-related developmental complications in the future. The owner elected humane euthanasia and a necropsy was performed to confirm the malformations. A diagnosis of complete urogenital and anorectal duplication was made via contrast computed tomography and necropsy in a 10-week-old intact female golden retriever. Clinical signs included smaller, soft, poorly formed, fecal material from both anal openings and urinary incontinence which had been present since birth. Advanced imaging of the patient was pursued, but surgical correction was declined. A necropsy was performed to further investigate the anatomical variations in this patient. These congenital abnormalities were reviewed in the veterinary literature and compared with human descriptions of similar abnormalities.A 10-week-old 9.6 kg sexually intact, female, golden retriever puppy was purposefully bred and born via cesarean section with ten other littermates . The female dog in question had two smaller than anatomically normal slit-like anal openings with haired skin coursing between the two distinct orifices. The referring veterinarian examined and explored the female puppy's abnormality after birth and ruled out a persistent anal membrane. No further treatment or surgery was performed since the female puppy did not appear to have difficulty with defecation. The dog was then referred to the University of Florida Small Animal Hospital for further evaluation and exploration of surgical treatment options.On presentation, the dog was bright, alert, and in good condition, with a body condition score of 5/9 and normal vital signs. The owner reported the patient defecated through both anal openings without tenesmus and noted that the fecal material was mostly soft and of poorly formed consistency. During the physical examination, the dog became excited and dribbled urine in several small spots. Further discussion with the owner revealed that she frequently found urine on the floor but did not recognize this as a sign of incontinence. On physical examination, the left anal orifice was situated 3-4 mm off midline, was smaller in diameter, and had a slit-like appearance compared to the right anal opening, which was 1-2 mm to the right of midline . On rectAbdominal radiography was obtained to rule out possible orthopedic and vertebral anomalies and to examine the abdominal contents. Abdominal radiography revealed a normal sized stomach containing a small amount of gas and soft tissue material. The small intestines were normal in diameter and had a normal distribution within the abdomen, and a few segments had a small amount of central contiguous gas. The colon was normal in size and contained a small amount of fecal material and gas that extended caudally within the rectum to the perineal margin. Serosal detail was normal with no other abdominal abnormalities noted. No orthopedic or vertebrae malformations were noted. The visible portion of the thorax was within normal limits. Based on these findings, contrast computed tomography (CTA) was recommended to further define the congenital anomalies.2 were monitored throughout the procedure. Lidocaine was administered to the patient during the procedure at 1.66 mg/kg intravenously.A packed cell volume: 32% (reference range 37-54%), total protein: 4.6 g/dL (reference range 5.4-7.1 g/dL), and AZOstick  were performed prior to induction of anesthesia. Premedication and induction of general anesthesia were performed with 1 mg of butorphanol intravenously (0.1 mg/kg), and 30 mg of Propofol (3.13 mg/kg) plus 15 mg ketamine (1.56 mg/kg) respectively and the patient was maintained on isoflurane inhalant ranging from 1 to 2% and oxygen at 1L/min for the duration of the CTA imaging series. Heart rate, electrocardiogram, indirect oscillometric blood pressure, oxygen saturation (by means of pulse oximetry), and end-tidal COAdvanced imaging, by computed tomography  of the abdomen and pelvis, was performed with intravenous administration of a nonionic, iodinated contrast medium, Iohexol (300mg I/ml), for a total of 4.8 g (500 mg/kg). Images were obtained after contrast administration during both arterial and venous phases. Helical CT images were obtained in a volume data set and were reconstructed in soft tissue, bone, and lung algorithms and then reformatted in transverse, dorsal, and sagittal planes.A vaginocystourethrogram was performed using two 10 French Foley catheters (one in each vulva), using approximately 15 mL of nonionic, iodinated contrast medium (Iohexol 300 mg I/ml) in each catheter to confirm complete or incomplete duplication as well as establish whether communication was present. Subsequently, a retrograde colonogram was performed using two 10 French red rubber catheters , secured by purse string sutures of 2-0 Nylon (Ethilon®) suture. Approximately 30 mL of barium sulfate paste positive contrast medium was administered in each red rubber catheter. The dog was rescanned, and images were acquired after vaginocystourethrogram and retrograde colonogram.CT images and abdominal radiographs were reviewed and interpreted by a board-certified radiologist. A vertically oriented soft tissue septum was present and measured 0.37 cm thick and 4.8 cm in length, which extended from the anus to the caudal rectum diverging the positive contrast medium laterally. The contrast then joined again cranially at the level of the second caudal vertebra . The vulSurgical options for correction of the anal abnormalities were presented to the owner. However, due to the urogenital findings and concurrent urinary incontinence, the owner instead elected humane euthanasia. The dog was euthanized with 3 ml of sodium pentobarbital (390mg/ml) given intravenously at 122 mg/kg.A complete necropsy was performed by a board-certified pathologist (MJD). The urinary bladder was abnormal when viewed externally with a cranial-to-caudal oriented central depression. Internally, the urinary bladder was completely bifurcated by a soft tissue septum extending from the apex to the trigone. The trigone had separate urethras exiting in each urinary bladder chamber. The urethras each extended approximately 4.5 cm caudally and then entered separate duplicated vaginas, each with a separate vaginal canal and opening to the outside of the body . The uteThis case represents a unique group of anatomic anomalies not previously reported in the dog. Reports in the veterinary literature are limited, encompassing only four canine cases –4 and onOf the four canine cases, three were not treated surgically. A seven-week-old mixed breed puppy was found to have partial sacral duplication, hemivertebra, and duplication of the bladder, descending colon, and rectum . A nine-The only reported case of colonic duplication surgically repaired in veterinary medicine was a six-month-old intact female, Boston terrier . This doThere is one report of urinary bladder duplication in a cat with urine soaked perineum and was thought to have ectopic ureters antemortem. The abnormalities were diagnosed at necropsy after presenting dead on arrival 35 months after ovariohysterectomy [To the authors' knowledge, this case represents the most extensive colonic and urogenital malformations reported in a dog in veterinary medicine. There are numerous reports in the human literature describing rectal or colonic duplications –14. MostDuplication of the colon can be associated with complete duplication of the urinary bladder and the urethra, which is a rare anomaly typically reported in children within the first few months of life , 15. EmbIn the human literature, Kottra and Dodds reported a classification, consisting of two types, for colonic duplication based on the work of Smith , 16. A tUrinary or urogenital malformations in humans come in two forms: those patients with clinical signs that present early in life, and others that remain asymptomatic for years . Human pIn addition to the colonic and urogenital malformations, the dog described in this case report also lacked a caudal mesenteric artery. The caudal mesenteric artery provides blood supply to the distal colon and rectum. Variations of the caudal mesenteric artery are previously described in dogs. In a recent study of five normal dogs, the caudal mesenteric artery branched off the aorta in a leftward direction in three dogs (60%) and in a rightward direction in two dogs (40%) .In our patient, the urogenital and colonic malformations were detected early in life. Both surgical and medical treatment options were discussed with the owner. Surgical repair options were limited and carried a high risk of morbidity without potential benefit to the patient. The owner raised concerns about the dog's quality of life and concern for multiple surgical procedures, so humane euthanasia of the dog was elected. This case represents the intricate process of embryonic development, the critical utilization of cross-sectional imaging with regard to surgical planning, and the utility of correlating cross-sectional imaging which verify the anatomic pathology findings.


Congenital intracranial tumors are very rare and account for less than 2% of all childhood brain tumors. Teratomas constitute about one third to one half of these, predominantly located midline in the supratentorial region. Posterior fossa location rarely occurs and, based on the cases reported in the literature, commonly has a poor prognosis.A newborn female, diagnosed prenatally with hydrocephalus, is presented at birth with increasing head circumference and Parinaud’s syndrome. Magnetic resonance imaging scans demonstrated a huge posterior fossa tumor with obstructive hydrocephalus. At surgery, through a suboccipital craniotomy, complete excision was achieved of a histological-proven immature teratoma. The infant received adjuvant chemotherapy for 1 year. She had normal neurological development and remained tumor-free through her 20-year follow-up.The authors report this rare case of congenital posterior fossa teratoma with long-term outcome, and the literature is reviewed. Congenital intracranial tumors are a rare occurrence, accounting for only 0.5–1.5% of all childhood brain tumors , 3. TeraThe Authors report this rare case of a posterior fossa congenital teratoma presented with Parinaud’s syndrome with long-term survival and review the literature.A newborn female of an uneventful pregnancy of a 40-year-old woman was delivered on 1 January 1999 via cesarean section (CS) at full term, because of previous CS. She is the sixth child of non-consanguineous Saudi Arabian parents who were originally from Gizan. During her gestational period, routine fetal ultrasonographic (US) scans at the 34th week reported mild ventriculomegaly without mention of any associated brain tumor.At birth, her Apgar score was 8 and 10, and her weight was 3150 g. Her head circumference after birth was in the 90th percentile, and the anterior fontanel was 20 × 20 mm and soft. The initial neurological exam was normal, apart from a squint, and her parents were reassured.At the age of 3 days, her mother brought her to a polyclinic because of poor feeding. She was reassured, and a change of milk formula was satisfactory for 1 week. Because of her recurrent vomiting and irritability, a computed tomography (CT) scan was performed, which reported a large posterior fossa tumor with obstructive hydrocephalus.On admission at 20 days old to King Abdulaziz University Hospital in Jeddah, she presented with frequent vomiting, poor feeding, and increasing head circumference. The general physical exam revealed an irritable and emaciated baby in the second percentile of weight for her age. Head circumference was 45 cm with visibly dilated scalp veins and bulging anterior fontanel. Neurological exam demonstrated a conscious baby with spontaneous movement of her upper and lower extremities with mild spasticity. Cranial nerves exam was uneventful apart from Parinaud’s syndrome Fig. . PupillaThrough a midline suboccipital craniotomy, complete resection of the tumor was achieved. During surgery, an encapsulated solid vascular tumor occupying the fourth ventricle was encountered with diverse areas of soft and firm consistency. The baby remained stable hemodynamically throughout the surgery and received a total of 250 ccs packed RBC blood. During the operation, cerebrospinal fluid (CSF) was obtained for analysis, which was within normal limits for chemistry and cell counts. CSF cytology and tumor markers studies, human chorionic gonadotropin, and alpha-fetoprotein were negative. Histopathological examination of the specimen demonstrated a variety of tissues from all three germ layers, including immature as well as mature elements. The predominant tissue was neuroectodermal, in the form of neuroepithelial rosettes and tubules, resembling neuroblastoma. Mesodermally derived immature cartilage and primitive stroma were also seen, and endodermally derived respiratory and enteric epithelium was present in the form of cystic structures Fig. . The diaAt the first year anniversary follow-up, she had a mild developmental delay with a slight improvement of Parinaud’s syndrome. Follow-up MRI scans at the fifth year of age revealed the stable size of the ventricles with no tumor recurrence Fig. . The patPrimary brain tumors form 20% of pediatric neoplasms and are the second most prevalent cancer of childhood, after leukemia . TeratomThe incidence of intracranial teratoma is 0.5% of all brain tumors and 2 to 4% of intracranial tumors in the pediatric age group –11. ThisCommon presenting symptoms and signs of such tumors include symptoms related to the outflow obstruction of cerebrospinal fluid resulting in a headache, vomiting, and papilledema Table  4, 13]., 13.4, 1Furthermore, structural compression of the tectal plate by the tumor generates abnormal forces resulting in either brainstem ischemia or neuronal distortion before the development of hydrocephalus. Abnormal development of the tectal plate nuclei can be another cause of such syndrome in this patient population. This can be illustrated in our current case: partial improvement of Parinaud’s syndrome resulted after successful decompression, and no hemorrhage or infarction within the brainstem in the postoperative MRI scans suggests that tectal plate neurons may have been incompletely developed.In a patient with congenital cerebellar teratoma, diagnosis may be prenatal or postnatal . PrenataOnce the diagnosis of posterior fossa tumor is confirmed, the treatment of choice is complete excision of the lesion . AfterwaPoor survival of infants with congenital intracranial teratoma has been shown; a large number die prenatally and the rest shortly after birth . In theiCongenital posterior fossa teratoma is a rare tumor that presents with significant large size and massive obstructive hydrocephalus. Early surgical resection is recommended with adequate perioperative care. Regular clinical and radiological follow-up is essential to assess the child’s neurological function and watch for early recurrence. Despite insufficient data in the literature about factors that favor a good outcome for posterior teratoma, we note that complete surgical resection is a significant prognosticator.


Nevus unius lateris is a rare congenital hamartoma derived from the ectoderm, considered to be a systematized verrucous variant of an epidermal nevus. Due to its extensive unilateral distribution, it is frequently associated with neurological, musculoskeletal, auditory, and visual abnormalities. A case report of a 25-year-old female patient with a diagnosis of nevus unius lateris without associated comorbidities is presented. Epidermal verrucous nevus is a common clinical finding; however, its generalized linear distribution known as nevus unius lateris is uncommon. There have been approximately 200 cases reported  worldwidA 25-year-old female patient of Indian descent presented with verrucous hyperpigmented neoformations in the right hemibody at the level of the trunk, abdomen, back, genitals, groin, and leg, sparing the face, neck, and mucous membranes Figures -2. The lEpidermal verrucous nevus has an estimated prevalence in the general population of one in a thousand; however, its variant nevus unius lateris represents only 0.01 percent of this total . Due to Nevus unius lateris as an isolated dermatosis has rarely been described. It is a rare congenital dysembroplasia, which in most cases is associated with epilepsy, growth retardation, brain tumors and visual and auditory disturbances, making early diagnosis crucial. To the best of our knowledge, only two cases like this one have been previously reported, neither of which had a previous family history. Therefore, it is important to state that not all cases have a poor clinical prognosis and there can be a pattern of heritability, which had not previously been documented. Nonetheless, X-rays, computed tomography, and blood work should be done at birth and later in life to rule out associated disorders.


Introduction: Sinonasal schwannoma accounts for only 4% of head and neck nerve sheath tumors. Schwannoma of the nasal septum is still the rarest, with only few cases previously reported.Case presentation: This study presents the case of a 64-year-old Saudi female of the Eastern Province who presented a several-month history of unilateral progressive nasal obstruction and recurrent episodes of epistaxis. The diagnosis of nasal septal schwannoma was made on the basis of a histopathological examination. The case was managed successfully by endoscopic endonasal excision with no postoperative recurrence.Discussion: Nasal septal schwannoma is rare and has only 32 reported cases. Diagnosis primarily depends on histopathological examination. Endoscopic endonasal excision of the mass is considered the ideal treatment for such pathology.Conclusion: Schwannoma of the nasal septum is rare. However, it should be included as a differential diagnosis in a patient presenting with a unilateral nasal mass. The most common site of schwannoma in the head and neck region is the eighth cranial nerve (vestibulocochlear nerve). Other sites include the scalp, face, parotid gland, oral cavity, pharynx, larynx, and trachea . Sinonas2A 64-year-old Saudi female of the Eastern Province was referred to our institute with a several-month history of unilateral progressive nasal obstruction and recurrent episodes of epistaxis. There was no history of anosmia, nasal discharge, nasal congestion, sneezing, facial pain, or headache. Her past medical history included a known case of hypertension and diabetes. Her family history was otherwise unremarkable.A rigid endoscopic examination of the nose showed a large, left-sided nasal polypoidal mass with smooth overlying mucosa that occluded the whole left nasal cavity . The remA CT scan of the paranasal sinuses with contrast showed a left anterior lobulated nasal mass of undetermined origin at the level of the cartilaginous part of the nasal septum with mild heterogeneous post IV contrast enhancement and bone remodeling. There was no extension to the paranasal sinuses, and no obvious bone invasion was observed .Fig. 2CTEndonasal endoscopic excision of the left nasal cavity mass was performed under general anesthesia. Intraoperative findings demonstrated a semifirm vascular mass attached to the anterior face of the middle turbinate and septum at the same level. The mass was excised completely from its attachment, and the pedicle was cauterized. Histological examination of the mass revealed three fragments of soft to firm tan-gray tissue, the largest measuring 1.8 × 1.6 × 0.6 cm, and the smallest measuring 0.8 × 0.6 × 0.5 cm . The lesThe patient has received regular follow-up care for three years with no recurrence.3Nasal septal schwannoma is rare, and only 32 cases have been reported in the literature . SchwannThe differential diagnosis for unilateral nasal mass includes nasal polyposis 22%), antrochoanal polyps (19%), chronic rhinosinusitis (13%), concha bullosa (11%), inverted papilloma (6%), and retention cysts (6%). Other less common potential causes include fibrous dysplasia, mucocele, lymphoma, schwannoma, ameloblastoma, pleomorphic adenoma, myxoma, and squamous cell carcinoma 2%, antro.CT scan findings are not specific for a schwannoma diagnosis. However, CT is helpful in evaluating the tumor’s origin and extent. MRI is favored over CT in differentiating tumors from inflammatory disorders and normal tissue; additionally, MRI provides information regarding intracranial invasion of the tumor . MRI hasBecause imaging is non-specific for schwannoma, nasal septal schwannoma diagnosis primarily depends on biopsy or complete excision of the mass. Macroscopically, schwannomas appear as well circumscribed, encapsulated, cystic masses that are connected to the nerve tissue. Microscopically, schwannomas are classified into two types: Antoni A and Antoni B patterns. Antoni A patterns are composed of spindle cells organized as cellular areas with nuclear palisading . Antoni The preferred treatment for nasal septal schwannoma is surgical resection of the mass, which may be achieved by lateral rhinotomy or endoscopic endonasal surgery . The end4Schwannoma arising from the nasal septum is rare. However, it should be included as a differential diagnosis in a patient presenting with a unilateral nasal mass. Histopathological examination is the gold standard test for diagnosis. Furthermore, endoscopic endonasal excision of the mass is the ideal treatment because it is a minimally invasive approach with improved visualization, lower morbidities, shorter hospital stays and external scar avoidance.There are no conflicts of interest.None.Ethical approval is exempted for the case report at our institution.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.Wejdan Alrasheed: manuscript draft and final edits.Ali Almomen: operating surgeon, data analysis and interpretation and critical revision of the manuscript.Abdulrahman Alkhatib: data collection and literature review.Not applicable.The corresponding author.Not commissioned, externally peer-reviewed.


A pancreaticoduodenal artery aneurysm (PDAA) occurring in close association with median arcuate ligament syndrome (MALS) is rare. A surgical procedure, such as median arcuate ligament (MAL) release, should be considered in such cases, but the operative criteria remain unknown. In this study, we reported an extremely rare case of PDAA with periarteritis nodosa (PAN) and MALS.A 60-year-old man was transferred to our department with sudden onset of abdominal pain. We initially diagnosed his condition as a PDAA rupture with MALS based on enhanced computed tomography (CT). We promptly performed transcatheter arterial embolization (TAE) of PDAA, and the angiogram showed stagnant contrast agent in the celiac trunk, indicating total celiac artery occlusion. Follow-up enhanced CT three weeks after the first TAE clearly demonstrated newly formed, multiple aneurysms in the pancreaticoduodenal arcade and the hepatic artery. These findings indicated a systemic disorder, such as PAN or segmental arterial mediolysis, as the underlying cause. Therefore, we started corticosteroid therapy and performed diagnostic angiography to clarify the celiac artery’s patency. Contrary to the initial angiography, the second angiography showed sustained blood flow in the celiac artery. Nevertheless, we performed both extrinsic MAL release and consecutive TAE because of the risk of multiple aneurysms rupturing due to an uncontrolled systemic disorder and consequent hepatic ischemia. The patient had no episode of recurrence until one year of follow-up.It is important to evaluate risk for hemodynamically unstable events to decide the best treatment strategy for MALS. Median arcuate ligament syndrome (MALS) is caused by a stenosis or occlusion of the celiac trunk by the median arcuate ligament (MAL). In response to the high arterial resistance caused by stenosis or celiac artery occlusion in MALS, pancreaticoduodenal artery aneurysm (PDAA) frequently occurs because of increased collateral blood flow through the pancreaticoduodenal arcade from the superior mesenteric artery (SMA).Depending on the aneurysm’s location, extrinsic MAL release from the celiac trunk has to be considered before any intervention because liver ischemia can sometimes occur as a complication of embolizing aneurysms. However, PDAA caused by MALS is such a rare disease that no therapeutic guideline or criteria for MAL release has been established.Here, we report a rare case of ruptured PDAA with MALS, in which laparoscopic MAL release was effective despite maintenance of substantial celiac blood flow on preoperative angiography. We emphasized the importance of establishing therapeutic criteria for PDAAs with MALS.A 60-year-old man was transferred to our department with sudden onset of abdominal pain. He had a past medical history of duodenal ulcer and paroxysmal atrial fibrillation, and had taken rivaroxaban. He had no allergy and no family medical history.2), 100% at 6 L/min of oxygen by reservoir mask. He complained of sustained upper quadrant pain with abdominal guarding. Initial enhanced computed tomography (CT) demonstrated extravasation from the posterior inferior pancreaticoduodenal artery and celiac trunk stenosis. Thereafter, we diagnosed him with PDAA rupture due to MALS.Physical examination showed initial findings of Glasgow coma scale, E4V5M6; blood pressure, 85/66 mmHg; respiratory rate, 25 /min; and peripheral oxygen saturation  was performed Fig. . The PIPThe patient was followed-up with enhanced CT every week. Follow-up CT on day 21 after admission  was not controlled. Therefore, we deduced that unless MAL release was performed before pancreaticoduodenal arcade TAE, an aneurysmal rupture may lead to the same vasospasm and, possibly, fatal liver ischemia. Extrinsic MAL release was a beneficial and safe strategy in this case.There is no evidence on the necessary period for sufficient collateral blood flow to develop. PDAA is so rare that definitive criteria for MAL release are not yet available. To delineate these criteria, it may be necessary to consider the relationship between collateral blood flow occlusion and liver ischemia, including the growth speed of sufficient collaterals. This case strongly suggested that extrinsic MAL release must be considered in patients at risk for hemodynamic instability. Conversely, some have reported that extrinsic MAL release may be unnecessary. Sgroi et al. reported three cases without celiac artery revascularization, stating that revascularization may be unnecessary in asymptomatic patients . ShibataThere is also insufficient evidence with regard to long-term outcome of celiac trunk revascularization with both extrinsic ligament release and stenting; therefore, we chose the former strategy considering our experiences in this case.In conclusion, it is important to evaluate risk for hemodynamically unstable events to decide the best treatment strategy for MALS.


Dear Editor,A 44-year-old patient presented with axial sensorimotor deficit, dating backapproximately 10 days. The history was significant for diabetes, alcoholism, andcognitive impairment, making it difficult to assess the recent history and symptoms. Thepatient was submitted to brain magnetic resonance imaging (MRI) with T2-weighted imaging(T2WI), fluid-attenuated inversion recovery (FLAIR) sequences, susceptibility weightedimaging, and diffusion-weighted imaging (DWI), as well as T1-weighted imaging (T1WI),before and after intravenous gadolinium administration, in the axial, sagittal, andcoronal planes. In the right postcentral gyrus, MRI revealed a cortical lesion, whichshowed a hyperintense signal on FLAIR image . The les. Intravascularenhancement, although not specific, is considered a sign of ischemia on conventionalMRI. Contrast enhancement in the central nervous system is the result of a combinationof disruption of the blood-brain barrier, high vascularity, and contrast leakage intothe lymphatic system(3-6). After one week, infarctsshow parenchymal enhancement, due to breakdown of the blood-brainbarrier(7).Diabetes mellitus is a well-recognized risk factor for ischemic stroke, which is aleading cause of death and disability. MRI is quite sensitive in detecting ischemicchanges. T2WI is more sensitive than is T1WI, and T1WI after gadolinium administrationcan provide valuable information for the accurate diagnosis. In addition, because ofpseudonormalization, subacute infarcts may not show restricted diffusion on DWI.New imaging techniques, such as DWI and perfusion-weighted imaging, have increased theaccuracy of the diagnosis of acute cerebral ischemia, although there are some cases inwhich it cannot be distinguished from other entities(10).This is exactly what occurred in the case presented here, in which DWIpseudonormalization did not help reveal the subacute cortical infarct. When a subacutecortical infarct is suspected, delayed contrast-enhanced FLAIR imaging is the bestchoice for demonstrating the lesion and for differentiating it from an older lesion withgliosis.FLAIR is highly sensitive for the detection of ischemic lesions. Although it isconsidered to be heavily T2-weighted, rendering cerebrospinal fluid as dark, it alsoshows mild contrast enhancement on T1WI, which is responsible for the increasedconspicuity of gadolinium enhancement. Pathologic conditions that present contrastenhancement on T1WI usually show marked enhancement on contrast-enhancedFLAIR


Currarino triad is a rare syndrome that may be occasionally encountered during managing cases of anorectal anomalies. The triad consists of anorectal anomaly, sacral bony defect, and a presacral mass. It may be familial or sporadic, with a reported female predominance. Identification of the characteristic notched sacrum  in plain X-ray (anteroposterior view) is considered the key for the diagnosis; however, not infrequently, this radiological sign is overlooked, especially with a small sacral defect.Excision of the presacral cyst is usually performed concomitantly during anorectoplasty. The prone position is the standard approach for posterior sagittal anorectoplasty (PSARP) in males; however, in females, the supine position can be used as an alternative . In this case report, excision of the presacral cyst took place in two steps: the first excision during the PSARP procedure in the prone position, and a second operation in the supine lithotomy position to remove a residual component of the lesion that was missed during the primary operation. It was clear that the supine lithotomy position provided better access to explore the presacral space than the prone position, especially with a deeply located cyst as in our case. The role of magnetic resonance imaging (MRI) in the identification of the exact nature and extent of the lesion before surgery is crucial and should be performed in all cases. Currarino triad is a rare congenital disorder characterized by the presence of three components: anorectal anomaly, sacral vertebral defect, and a presacral mass .Early reports in the literature were described by KennedyA male patient presented at birth with anorectal malformation ; there At the age of 3 months, a high-pressure distal colostogram was performed as a preparatory step for a PSARP  procedure.At the time of operation, the mother revealed that her elder daughter (now 6 years old) underwent a similar operation. Revising the medical records of her daughter revealed a case of Currarino triad: anal stenosis (funnel anus), vertebral defect, and presacral cyst. Because of the reported autosomal dominant inheritance in cases of Currarino triad,With the patient in the prone position, a generous posterior sagittal incision was made for mobilization of the anorectum and exploration of the presacral space. We started as usual by dissection of the fascia around the posterior and the lateral aspects of the rectal pouch. A small midline incision was made in the posterior wall of the rectum to inspect for any missed communication with the urinary tract and to guide the dissection during separation of the anterior rectal wall from the intimately adherent urinary tract. After mobilization of the anorectum, a retrorectal cyst (∼2 cm in diameter) was found and excised. Then, the steps of anorectoplasty were completed as usual by reconstruction of the levator ani and striated muscle complex around the neorectum.Postoperative recovery was uneventful. Histopathological examination of excised specimen revealed fibromuscular tissue showing multiple clefts lined by stratified squamous epithelium and entrapped transitional epithelial islands. These histopathological findings go with the diagnosis of a developmental cyst associating with Currarino triad.Later, a postoperative pelvic MRI was ordered that revealed residual presacral cystic lesion , with tThe patient was seen at follow-up 3 months after the closure of colostomy. He defecates spontaneously once every 1 to 2 days. He is still only 19 months old and therefore below the age of voluntary bowel control. However, the mother was informed about the possibility of constipation, problems with fecal continence, and the need for long-term follow-up. The mother has had a similar experience with her elder daughter who is now 6 years old and has voluntary bowel and urinary control. The parents (being the first-degree relatives of our case) were advised to perform screening by plain X-ray sacrum.Currarino triad is a rare syndrome that may be occasionally encountered during managing cases of anorectal anomalies. The triad consists of anorectal anomaly, sacral bony defect, and a presacral mass.MNX1(previouslyHLXB9) gene on chromosome 7q36. The disorder is an autosomal dominant genetic trait that has variable expressivity.MNX1gene have been identified in nearly all cases of familial Currarino's syndrome and in approximately 30% of patients with sporadic Currarino's syndrome.Currarino triad is caused by heterozygous mutation in the-ray due to a highly variable phenotype within most families.-ray should be referred to a surgeon for further investigations, including pelvic MRI for an occult presacral mass due to significant inter- and intrafamilial variability in expression.All affected families should be offered genetic counseling, as awareness of the hereditary nature of the disorder allows identification of asymptomatic heterozygotes and patients at risk, which leads to better planning of pregnancies and appropriate management of affected patients. All first-degree relatives of patients with Currarino triad should be screened by pelvic XThe presacral mass in Currarino triad may be a lipoma, lipomyelomeningocele, or some sort of developmental cyst.Excision of the presacral cyst is usually performed concomitantly during anorectoplasty. The prone position is the standard approach for PSARP in males;There was some delay initially in completing the diagnosis for this case; however, the management was completed with a satisfactory outcome. Lastly, the prognosis for continence may be generally good for this case owing to the absence of associated spinal cord anomalies.


Inverted papilloma (IP) of the middle ear as a primary lesion or as an extension of a sinonasal papilloma, is extremely rare. Only 23 cases of primary inverted papilloma of the middle ear have been reported in the literature. They are locally aggressive tumours, with a high rate of recurrence and associated malignancy. We present a rare case of a 59-year-old man presenting with unilateral otorrhoea, tinnitus, and hearing loss. Otoscopy revealed posterior perforation filled by irregular budding neoformation. The temporal CT scan showed tissue filling the tympanic cavity as well as the mastoid antrum without bone lysis. The patient underwent limited tympanoplasty. An intraoperative biopsy of polypoid tissue filling the tymapanic cavity was made and histopathology showed an IP. A recurrence occurred 4 months after surgery. We performed open tympanoplasty allowing complete resection of the lesions, with no recurrence after a follow-up of 30 months. Inverted Papilloma (IP) is a benign tumor that develops primarily in the sinonasal tract. Localization in the middle ear, regardless of primary or extension of a sinonasal IP, is exceptional. Only 23 cases of primary inverted papilloma have been reported in the literature -6. From A 59-year-old patient consulted us for hearing loss, tinnitus and left otorrhea for one year. The patient had no dizziness or otalgia. He did not have a history of sinonasal pathology or surgery. Otoscopy revealed posterior perforation filled by irregular budding neoformation. The latter bleeds on contact. The contralateral ear was healthy. The rhinological examination was normal. The audiogram was in favor of a left hearing deafness of 50 db. The temporal CT scan showed tissue filling the tympanic cavity as well as the mastoid antrum without bone lysis . Magnetiet al in 1987 and since then only 45 cases have been published in the literature [Papillomas are rare benign tumors arising at the level of the Schneiderian membrane that covers the sinonasal tract. They are still called schneiderian papillomas. There are 3 types of papilloma: inverted papilloma is the most common 62%) followed by exophytic papilloma (32%) and oncocytic (6%). The histological diagnosis of inverted papilloma is based on the discovery of epithelial masses invaginated in the underlying chorion: this typical aspect is at the origin of the term “inverted” attributed to this tumor. The inverted papilloma of the middle ear is exceptional. It was first described by Ston terature . Three t2% followHearing loss and otorrhea are the most common symptoms. Tinnitus, otalgia, fullness of the ear, headache, and facial palsy are rare. Three otoscopic aspects have been described in the literature. It may be a complete tympanum but is repressed by a retro-tympanic neoformation. The second aspect is that of a tympanic perforation with a fund that is budding. The third aspect is that of an irregular and bleeding neoformation that fills the bottom of the external auditory canal. Some differential diagnoses may be referred to as middle ear adenoma, paraganglioma, cholesteatoma and endolymphatic sac adenocarcinoma. Only the histological examination that confirms the diagnosis of IP. IPs are characterized by a high risk of local recurrence after surgery. The recurrence rate is higher than in the sinonasal localization. This can be explained by the fact that complete surgical excision is more difficult in the middle ear than in large sinonasal cavities. Similarly, the rate of malignant transformation of middle ear IP is greater than that in sinonasal locations , 10. In The middle ear IP is extremely rare. The clinical and radiological signs are not specific. Only the histological examination that confirms the diagnosis. It is characterized by a high rate of recidivism and malignant transformation. The treatment of middle ear IP is surgical but it is difficult to define a standard surgical technique. It is important to have a large and complete excision to avoid recurrence.The authors declare no competing interests.


Retained surgical items continue to occur despite widespread implementation of prevention systems such as the surgical count, which has limited utility owing to its reliance on human performance. The most important risk factors for these events are poor communication in the operating room and inconsistent adherence to protocol. New technologies show efficacy in preventing retained surgical items and partially mitigating the poor reliability of the manual count. Additionally, efforts to address systemic and environmental sources of error have demonstrated success in reducing the incidence of retained surgical items. Here, we present the surprising case of a patient with a retained surgical sponge presenting as a soft tissue mass four decades after his surgery. Retained surgical item (RSI) is defined as any sponge, tool, device, or other object left inside a patient following wound closure at the end of an operation. The Joint Commission considers RSIs sentinel events that require immediate investigation, root cause analysis, and response. The National Quality Forum designates them surgical “never events,” or serious and preventable adverse events that should not occur if standards of care are upheld. These events have undisputed consequences in terms of physical harm to patients, cost of reoperation and readmission, and legal ramifications . DespiteAn 87-year-old male was referred for surgery by his primary care physician due to a mass on his right hip that had been enlarging over the preceding three months. His surgical history was pertinent for a fusion of cervical vertebrae C3 and C4 approximately 45 years prior, although he did not recall the exact year, during which a bone graft was harvested from his right iliac crest. One week after his fusion, he reported pain, bleeding, and purulent drainage from the site of the graft. He was treated with antibiotics as an outpatient and remained asymptomatic until noticing a hip mass three months prior to presentation. A pelvic CT scan showed “radiopaque surgical material” surrounding a soft tissue mass Figures –3.On the operating table, an elliptical incision was made and the mass was excised to the level of the ilium, revealing a cavity in the bone from which two Raytec© surgical sponges were removed. Cultures were obtained, and the cavity thoroughly irrigated. His postoperative course was without complications. Cultures showed no organisms after 72 hours, and gross pathology revealed soft tissue with a foreign body reaction and hemorrhage , as wellRSIs may present as early as the day of surgery, with most discovered within weeks to months . RetentiReported risk factors for RSIs vary by source and span many domains. Those pertaining to the patient or procedure include high patient BMI, prolonged procedure duration, and emergency surgery . Of arguThe current mainstay of preventing RSIs is the surgical count, along with X-ray imaging in the case of unreconciled items. The count relies heavily on human performance and is thus prone to error, particularly in settings that compromise standard protocols, such as emergent surgery. To this point, several studies have found that a majority of RSIs occurred where the surgical count was declared to be “correct” or an incorrect count was never reconciled , 6. SuchThe aims of this report are two-fold: first, to share the case of an RSI with a markedly prolonged period of retention; and second, to encourage readers to revisit the circumstances that perpetuate RSIs and other “never events” and thoughtfully evaluate the efficacy of preventive efforts. Optimization of existing prevention methods, such as the surgical count, requires minimizing distractions in the operating room, clear communication, and consistent adherence to protocols, with augmentation from counting and detection technologies. The limitations of the count should provoke ongoing discussion about the nature of human error and the substrate on which it occurs. Though there is often inclination to assign individual blame, this is neither accurate nor useful. Human variability and the complex environment of the operating room are better suited to a systems model of error, wherein efforts turn toward understanding the circumstances that permit mistakes, optimizing the work environment to minimize them, and encouraging a culture of transparency .


Extrapleural hematoma is uncommon. However, according to the size of hematoma and/or the progression of anemia, surgical treatment to control bleeding might be necessary because a huge hematoma can cause ventilator and circulatory disturbances to press heart and lung. We present two unusual cases of huge extrapleural hematoma in an anticoagulated patient with no apparent history of trauma or otherwise traumatic episodes.Case 1: A 78-year-old man presented to our emergency department with pain in his right shoulder and disturbance of consciousness. He had no apparent history of trauma. Computed tomography (CT) of the chest revealed the presence of a huge lens-like encapsulated lesion measuring 220 × 90 mm in the right thoracic cavity. These findings all supported a diagnosis of extrapleural hematoma with hemothorax. Case 2: A 73-year-old man was brought to our hospital by ambulance after bruising his back in his house. CT of the chest revealed the presence of a huge lens-like encapsulated lesion measuring 230 × 70 mm in the left thoracic cavity. Hemorrhagic effusion was obtained by thoracocentesis, and the lesion was suspected of being a hematoma. In both two cases, we performed video-assisted thoracic surgery (VATS), which was minimally invasive and effective. These two patients were cured and discharged smoothly after surgery.We reported two rare cases of extrapleural hematoma. This disease requires close attention when it manifests in patients undergoing anticoagulation therapy. Regarding treatment, VATS was particularly effective in these cases. Extrapleural hematoma is uncommon. However, according to the size of hematoma and/or the progression of anemia, surgical treatment to control bleeding might be necessary because a huge hematoma can cause ventilator and circulatory disturbances to press heart and lung . Poyraz We herein report two cases of extrapleural hematoma and review the relevant literature.A 78-year-old man presented to our emergency department with pain in his right shoulder and disturbance of consciousness. He had no apparent history of trauma. His blood pressure was low (systolic blood pressure: 70 mmHg), oxygen saturation was 90%, and hemoglobin was 8.3 g/dl. His medical history included a giant vertebral artery aneurysm and coiling therapy performed 8 days ago, after which had been taking antiplatelet agents (aspirin and clopidogrel sulfate). Therefore, we considered that he was in hemorrhagic shock.Chest radiography revealed massive right hemothorax Fig. . ComputeWe performed video-assisted thoracic surgery (VATS). A huge hematoma was found in the extrapleural space Fig. , and aboA 73-year-old man was brought to our hospital by ambulance after bruising his back in his house. He complained of pain at his left back. His blood pressure was normal, oxygen saturation was 96%, and hemoglobin was 9.0 g/dl. His medical history included arteriosclerosis obliterans and bypass surgery, after which he had been taking an antiplatelet agent (aspirin).Chest radiography revealed massive left hemothorax Fig. . CT of tWe performed VATS. The intrathoracic findings were similar to those of case 1. A huge hematoma was found in the extrapleural space, and about half of it was unclotted. We located a bleeding intercostal vessel near the broken 10th rib. Therefore, we performed soft coagulation for this bleeding point and were able to stop the bleeding. We carefully search another bleeding point in thoracic cavity and confirm there was no bleeding using thoracoscopy before the chest was closed. His postoperative course was uneventful. The patient was discharged from our hospital on postoperative day 7.This report has three important implications: First, extrapleural hematoma is empirically rare. It is defined as the presence of blood in the extrapleural space between the parietal pleura and the endothoracic fascia and is created when the blood cannot escape into the pleural cavity because the parietal pleura is still intact . It has Second, VATS was minimally invasive and effective in both of our cases. Sumida et al. reported a case of a huge extrapleural hematoma in an anticoagulated patient without trauma  and suggOn that note, third, a soft coagulation system was useful for stopping bleeding. Sato reported that a soft coagulation system is a novel electrosurgical device that automatically maintains an output voltage below 190 V, resulting in pure coagulation without carbonization. Soft coagulation can be achieved with bipolar and monopolar devices in thoracic surgery. Bipolar scissors can be used to dissect pulmonary vessels safely and efficiently without damaging the vessel wall. Monopolar soft coagulation can be used to reduce bullous changes in the lung and stop air leakage from the lung parenchyma or bleeding from pulmonary vessels . The bleWe reported two rare cases of extrapleural hematoma. This disease requires close attention when it manifests in patients undergoing anticoagulation therapy. In case 1, this patient had no apparent history of trauma. However, he had been taking antiplatelet agents. Therefore, we thought that the intercostal artery may have failed even with a slight impact. Regarding treatment, VATS was minimally invasive and particularly effective in these cases.


Papillary thyroid carcinoma (PTC) is a common neoplasm from follicular cells of the thyroid. We report a case of pheochromocytoma/paraganglioma and PTC with negative testing for common mutations. Pheochromocytoma/paraganglioma is a rare tumor from neuroendocrine cells. 1/3 32-year-old male with incidental liver mass during laparoscopy for acute appendicitis. His symptoms included abdominal pain and profuse axillary hyperhidrosis. MRI showed an 11x12x14 cm cystic and solid mass in right adrenal gland, and 3.4x2.9x3.8 cm mass in porta hepatis. Urine metanephrines was elevated. After preoperative alpha-blockade, patient underwent total right adrenalectomy. Pathology report confirmed diagnosis of pheochromocytoma. According to the Grading system for Adrenal Pheochromocytoma and Paraganglioma (GAPP), tumor's score was 9, indicating poorly differentiated tumor. Ki67 index 5% and S100 were negative. Postoperatively, plasma free metanephrines normalized but plasma free normetanephrines remained elevated. Based on this biochemical profile, presence of paraganglioma was suspected. CT showed 4.2x3.5 cm round soft tissue mass in porta hepatis which increased in size from previous MRI. Simultaneously, PET scan identified a 1.5 cm thyroid mass. Calcitonin level was normal. Fine-needle aspiration was consistent with PTC. Resection of the mass and total thyroidectomy were performed with confirmation of paraganglioma S100 positive and PTC. Normetanephrines decreased to 283 (<148 pg/mL); free metanephrines remained normal. Gene mutation of EGLN1, FH, KIF1B, MEN1, NF1, RET, SDHAF2, SDHC, SDHD, TMEM127, VHL, and SDHA was negative. Whether paraganglioma/pheochromocytoma/PTC combination is coincidental or resulted from an underlying unknown mutation cannot be excluded. Increasing numbers of susceptibility genes have been identified, and approximately 1/3 SDHB mutations. SDH gene encodes for portions of mitochondrial complex II which is a tumor suppressor gene involved in the electron transport chain and the tricarboxylic-acid (TCA) cycle. There are different subunit genes: SDHB, SDHC, SDHD, SDHAF2, and SDHA. Multiple paragangliomas have been identified in 66.9 percent of the SDHD mutation carriers; but malignant paragangliomas are more commonly seen in SDHB mutation carriers, 37.5 percent, as opposed to 3.1 percent of the SDHD, and none of the SDHC mutation carriers. [ SDHB, SDHD, VHL, and RET. Patients with clinical predictive features should undergo genetic counseling.Endocrine society clinical practice guideline recommended genetic testing using clinical driven diagnostic algorithms. They suggest testing for succinate dehydrogenase (SDH) mutations and that patients with metastatic disease should undergo testing forWe report a case of pheochromocytoma, paraganglioma, and PTC discovered subsequently with negative testing for most common mutations. Figures 32-year-old male without significant past medical history was found to have what appeared to be a liver mass during surgical exploration for an acute appendicitis. His symptoms preoperatively included intermittent abdominal pain and profuse episodic axillary hyperhidrosis. On examination, he was normotensive and did not show evidence of pallor, anxiety, or tachycardia. 24-hr urine metanephrine was 4339 (36-190 mcg/24 hr), normetanephrine 20025 (35-482 mcg/24 hr), and total metanephrines 24364 (116-695 mcg/24 hr). MRI of the abdomen showed an 11 x 12 x 14 cm mixed cystic and solid mass centered in the right adrenal gland and 3.4 x 2.9 x 3.8 cm mass in the region of the porta hepatis.After preoperative alpha-blockade patient underwent total right adrenalectomy. Pathology report confirmed the diagnosis of pheochromocytoma. According to the Grading system for Adrenal Pheochromocytoma and Paraganglioma (GAPP) tumor's score was 9 , indicatPostoperatively plasma free metanephrines had normalized at 31 (<57 pg/mL) but plasma free normetanephrine remained elevated at 1844 (<148 pg/mL). Based on this biochemical profile presence of paraganglioma was suspected. CT abdomen showed 4.2 x 3.5 cm round soft tissue mass in the region of the porta hepatis which slightly increased in size from previous MRI.Simultaneously, positron emission tomographic scan identified a 1.5 cm thyroid mass. Calcitonin level was normal. Fine-needle aspiration of this nodule was consistent with papillary thyroid carcinoma. Resection of the porta hepatis mass and total thyroidectomy were performed with subsequent confirmation of paraganglioma and PTC, respectively. On surgical pathology paraganglioma was S100 positive. After the resection, level of plasma free normetanephrine had decreased to 283 (<148 pg/mL), and free metanephrine remained normal. Genetic studies, which included gene sequence changes and deletion/duplications of EGLN1, FH, KIF1B, MAX, MEN1, NF1, RET, SDHAF2, SDHC, SDHB, SDHD, TMEM127, VHL, and SDHA, were negative.Pheochromocytomas are rare tumors, widely described due to their serious morbidity and mortality rates. Most are sporadic tumor but 30% , 5 are iGermline mutations are five times more common among young patient, less than 45 years old; additionally multifocal or metastatic presentation has been associated with highest prevalence for gene mutation, four times higher when the tumor is extradrenal [From our review of literature of the studies found with simultaneous pheochromocytoma/paraganglioma and papillary thyroid carcinoma, the most comprehensive one was a four-case series analysis. It consisted of four female patients, one of them with synchronous PTC and PGL; 2 had heterozygous germline variants in SDHB and the four of them had -79 T>C CDKN1B gene polymorphism (3 homozygous and 1 in heterozygous state) . This geWhether paraganglioma/pheochromocytoma/PTC combination in our case is incidental or results from an underlying genetic predisposition is difficult to ascertain. Presence of unknown mutation cannot be excluded. The accelerated growth in genetic knowledge should prompt identification of additional susceptibility genes to be added in our genotyping panel in cases with high suspicion of mutation involvement such as presentation with multiple neoplasia.


We present the case of a male with a history of nasal polyposis underwent bilateral nasosinusal endoscopic surgery. He went to the emergency department because of having behavioral changes and left frontal headache. An emergency CT showed nasal-sinus polyposis and several nodular lesions with a characteristic “ring” enhancement and perilesional edema. These findings were compatible facial mucocele complicated with rupture of the bone wall of the left frontal sinus and frontal abscess. Urgent surgery was performed, with left frontal craniectomy and drainage of the abscesses. Mucoceles are benign slowly growing lesions which can associate important complications. The most frequent are abscesses and the invasion of neighboring structures. It is very important to remember that frontal mucoceles can cause intracranial invasion when there is an erosion of the internal osseous table. A 79-year-old male with a history of nasal polyposis underwent bilateral nasosinusal endoscopic surgery.He went to the emergency department because of having behavioral changes from 20 days ago and left frontal headache for 1 month. Blood test showed mild leukocytosis without associated elevation of acute phase reactants. Urgent CT was requested with the aim of ruling out brain complications.There are multiple entities that can present characteristics similar to mucoceles. It is necessary to consider the following entities within their differential diagnosis: mucous retention cyst, inflammatory polyp, mycetoma, and the neopasias of the paranasal sinuses.CT showed several nodular lesions with a characteristic “ring” enhancement, accompanied by marked perilesional edema which caused mass effect . In addiStaphylococcusaureus. The post-surgical evolution was favorable and the patient was discharged 10 days later.Urgent surgery was performed, with left frontal craniectomy and drainage of the referred abscesses. Samples were taken for microbiological cultures, with result of positive Mucoceles are benign, slow-growing cystic lesions filled with mucus and lined with epithelium. They usually associate occupation and expansion of the paranasal sinus in which they originate. The formation of mucoceles in the paranasal sinuses is associated with obstruction of the sinus drainage ostium.As a general rule, all those pathologies that hinder or prevent sinus drainage have the potential to produce mucoceles. The entities most associated with their training are the inflammatory–infectious processes, the trauma and the surgery, being less frequent its association with fibro-osseous lesions of the facial mass.1 They can be located simultaneously in more than one sinus region or even be bilateral, although the latter happens rarely.1The most frequent location of the mucoceles are the frontal sinuses (60–67%), followed by the ethmoidal cells (20–25%), maxillary sinuses (5–10%) and sphenoid sinuses (<5%).Over time, the mucoid material retained inside the corresponding paranasal sinus causes an increase in sinus pressure and its bony walls turn thinner. Secondary, bone osteolysis and remodeling are produced. In advanced cases, the lesion may reach the locoregional structures (orbit and/or brain parenchyma). It may cause intracranial complications of infectious etiology.T11 weighted sequences and hypointense in T2. As the protein content increases, the signal will increase in the T1 weighted images and will decrease in T2.2Imaging tests play a fundamental role in its diagnosis, mainly CT and MRI. In CT, mucoceles usually have a homogeneous content of low attenuation However, the density may vary depending on the content of water and proteins, as with the intensity of signal in the MR. In latter stages, mucoceles with a high concentration of water will be hyperintense in 3 Nowadays, new techniques like perfusion CT (PCT) and functional MRI modalities let to study tumor physiology.Conventional CT and MRI provide anatomic information and rely on morphologic changes secondary to paranasal lesions or brain tumors. However, they provide very little information about tumor functional behavior.4,5 On the other hand, functional MRI modalities such as diffusion MRI, dynamic susceptibility perfusion contrast MRI and MR spectroscopy also have been applied to investigate paranasal/brain tumors.6 Furthermore, diffusion has been used to differentiate benign and malignant lesions, due to the apparent diffusion coefficient (ADC) values of tissues vary according to their patology and cellularity.7On the one hand, the most important advantage of PCT is the linear relationship between contrast concentration and attenuation in CT, it facilitates quantitative measurement of blood flow and volume flow, so it let to identify tumor angiogenesis.In spite of being benign diseases, the mucoceles have complications, being the most frequent the superinfection and the invasion by contiguity of neighboring structures. Therefore, the knowledge of its main manifestations, both clinical and radiological, will be crucial to reach an early diagnosis that allows guiding the therapeutic attitude, avoiding that the prognosis of a benign entity is overshadowed by the appearance of such complications.The clinical case that concerns us represents a combination of both entities: the invasion by contiguity of neighboring structures such as brain parenchyma and superinfection/abscess. The structures that are affected by neighborhood depend on the location of the sinus in which the mucocele originates. On the one hand, orbital invasion is more frequent in the ethmoid and frontal sinus. On the other hand, sphenoid mucoceles are more likely to invade the cavernous sinuses, carotid vessels, optic nerves and the anterior and/or medial cranial fossa. Special mention should be made of the frontal mucoceles, which due to their proximity to the anterior cerebral fossa can cause intracranial invasion. This fact occurs when there is an erosion of the internal osseous table, causing important intraparenchymal complications, most of them of an infectious nature: cerebral abscesses, epidural/subdural abscesses or meningitis. The case presented is a clear example of this assumption. In the context of a marked nasosinusal polyposis and longstanding frontal mucocele, a thinning and solution of continuity of the posterior bony wall of the left frontal sinus was developed, with the consequent opening of the mucoid material to the ipsilateral frontal lobe and subsequent superinfection.6 In addition, when a CT is performed, it is very important to pay attention to the bone window  are seen, secondary to intracranial invasion.Bone erosions in the sinus walls translate intracranial extension of the lesion.


Bronchogenic cysts are rare, congenital cysts originating from respiratory epithelium and typically found within the chest. Cutaneous bronchogenic cysts are exceedingly uncommon, with only 19 reported cases in the scapular region and almost exclusively occurring in male patients. Herein, we present the case of a female patient with recurrent cellulitis secondary to a bronchogenic cyst, which was diagnosed after surgical excision. We also provide a review of the literature to consolidate the current understanding of cutaneous scapular bronchogenic cysts. To our knowledge, this is the first such case reported from Canada. During normal embryologic development, the foregut divides into ventral and dorsal components, which go on to form the trachea and esophagus, respectively . As deveOur patient is a 5-year-old female who was referred for evaluation and management of recurrent episodes of cellulitis in the left scapular region. A small cystic lesion had first been noted at the age of 2 years. While this had initially been an asymptomatic, small lump that grew in size over time, it first became symptomatic when the patient was 4 years old. The lesion developed surrounding induration and erythema, as well as purulent drainage and tenderness. Over the next 1-year period, she went on to develop 3 such episodes of cellulitis. This was treated with a 10-day course of cephalexin by the patient's family physician, and it was due to these recurrent episodes of infection that we saw the patient in consultation.Physical examination revealed a playful and well-appearing 5-year-old female, weighing 24.8 kg and measuring 114.5 cm in height. Review of systems and cardiorespiratory examinations were unremarkable. On inspection of the left scapular region, approximately 7 × 5 cm area of cellulitis was noted, with a small opening and associated purulent drainage. The surrounding skin was tender to palpation, but there was no appreciable fluctuance. Thus, the initial working diagnosis was infected epidermoid cyst. An ultrasound of the affected area showed a complex cystic mass measuring 3.9 × 2.9 × 3.7 cm within the subcutaneous fat . Deeper Overall, bronchogenic cysts are quite rare and have a reported incidence of 1 in 42,000–68,000 , 11, 12.Scapular bronchogenic cysts are exceedingly uncommon, with 19 cases reported in the literature so far, and our case being the 20th . The patOur case is a unique presentation of a scapular bronchogenic cyst in a female patient as recurrent cellulitis. To our knowledge, this is the first such instance reported within Canada. Further research is required to elucidate the exact mechanisms underlying the formation of all bronchogenic cysts, and certainly, an index of suspicion should be reserved for this diagnosis when working up scapular lesions in children.


Lemmel syndrome is a rare and misdiagnosed cause of acute abdominal pain due to a juxtapapillary duodenal diverticulum causing mechanical obstruction of the common bile duct. Frequently, patients suffering from Lemmel syndrome have a history of recurrent access to the emergency room for acute abdominal pain referable to a biliopancreatic obstruction, in the absence of lithiasis nuclei or solid lesions at radiological examinations. Ultrasonography (US) may be helpful in evaluation of upstream dilatation of extra-/intra-hepatic biliary duct, but computed tomography (CT) is the reference imaging modality for the diagnosis of periampullary duodenal diverticula compressing the intrapancreatic portion of the common bile duct. Recognition of this entity is crucial for targeted, timely therapy avoiding mismanagement and therapeutic delay. The aim of this paper is to report CT imaging findings and our experience in two patients affected by Lemmel syndrome. PAD incidence increases with age, observed in around 10–20% of patients undergoing endoscopic retrograde cholangiopancreatography (ERCP), with no gender predilection. PAD is acquired extraluminal outpouching of the duodenal wall through “locusminoris resistance” located within 2–3 cm to ampulla of Vater. Even if usually asymptomatic, PAD can cause acute abdominal pain due to an extrinsic obstruction of common bile duct or pancreatic duct, miming a biliopancreatic colic. While acute abdomen resulting from common intra-abdominal pathology has been extensively covered in the medical literature as well as the role of cross-sectional imaging in depicting acute or subacute abdominal conditions, the role of imaging in the diagnosis of symptomatic duodenal diverticula is an underreported entity, mainly due to the absence of specific pathognomonic symptoms and signs.Lemmel syndrome, first described by Lemmel in 1934,Lemmel’s syndrome could be challenging to diagnose, but recognize this condition is crucial to avoid wrong treatment. The first step in diagnosis is the identification of PAD . For this reason, PAD is better characterized after oral and/or intravenous contrast material administration: oral contrast  could be helpful in evaluation of diverticulum size before and after administration, as diverticulum could double in maximum diameter after oral contrast and could reveal underlying extrinsic compression on biliary duct [PAD may appear as rounded collection of gas material situated along the medial wall of the junction of the second and third portions of the duodenum on unenhanced CT, sometimes filled with fluid and misdiagnosed as a pancreatic abscess, pancreatic pseudocyst, or as a metastatic lymph node.ary duct . IntraveA 79-year-old male with acute abdominal pain after having presented several self-limiting episodes of abdominal pain in the right hypochondrium and epigastrium, underwent US examination in the emergency department, with no significative findings. He was discharged 6 h later. After 2 days, he was readmitted to the emergency department for increased epigastric pain happened about an hour after lunch. As slight increase in total bilirubin was found in blood test, unenhanced CT scan of the abdomen was required. It demonstrated the presence of a PAD associated with common bile duct dilatation . No otheA 70-year-old female presenting with nausea, vomiting, weight loss, and slight jaundice was evaluated for acute cholecystitis. Laboratory values revealed that hemoglobin was normal, but bilirubin metabolites and inflammatory markers, including CRP, were elevated. US demonstrated dilated common bile duct with no signs referable to lithiasis. A contrast-enhanced CT scan was required to study in deep pancreatic head region.CT retrospectively revealed the presence of a 17-mm PAD  and confWe reported two cases of PAD related to Lemmel syndrome. Lemmel syndrome is an unrecognized cause of jaundice and/or common bile duct dilatation because more frequent and important causes (choledocholithiasis or pancreaticobiliary and periampullary tumors) are first depicted. Imaging is essential to correctly identify and diagnose Lemmel syndrome, as awareness of this condition may prevent mismanagement. CT scan is the reference imaging modality because majority of patients present to emergency department with acute abdominal pain. US could reveal biliary duct dilatation, but the inherent limited ability to examine bowel pathology results in non-depiction of the duodenal diverticula. PAD could be evaluated through CT, MRI, or ERCP, which could be also therapeutic.,5-7There are few data reported in literature about imaging related to Lemmel syndrome, mainly case reports evaluated with ERCP.et al. in their study on small bowel diverticular disease described three cases of symptomatic duodenal diverticula focusing on the clinical findings without imaging correlation. Abdominal cross-sectional imaging has been reported to be a useful diagnostic tool in defining duodenal pathology and overcoming the aforementioned difficulties of clinical and ultrasound examinations.Kouraklis et al. reported 10 patients, all presenting with acute abdomen and diagnosed with duodenal diverticula, documented with MDCT in eight cases, in which they focused on the importance of both axial and coronal plane in demonstrating the presence of the diverticula.A study of Perdikakis et al. presented a case of Lemmel’s syndrome with a large PAD causing obstructive jaundice and mimicking periampullary tumor: in this case, an endoscopic ultrasound was performed, revealing a 2 cm diameter duodenal diverticulum with a small flange that increased in size (more than 50%) on instillation of water and resulted in obvious compression of both the biliary tract and main pancreatic duct, confirming the diagnosis of Lemmel’s syndrome. We also demonstrated that after oral contrast administration, PAD could significative increase in size, causing dilatation of common bile duct, even if oral hyperdense contrast media could hinder small CBD stones.Rouet PAD is best demonstrated using a side-viewing endoscope, but it remains an invasive imaging modality which could reveal that the distal portion of the common bile duct is laterally compressed by the PAD.Patients who do not undergo ERCP or endoscopic examination may benefit from the ability of MR and magnetic resonance cholangiopancreatography (MRCP) to detect PAD, enabling determination of the etiology of clinical symptoms and guidance for appropriate management.,10 Conservative management consists of nasogastric decompression and wide spectrum antibiotic coverage in cases of perforation. Therefore, duodenal diverticula are usually untreated unless complications occur. If conservative treatment options fail, surgical transduodenal diverticulectomy is usually performed.Although duodenal diverticula can be found in up to 22% of the population, only 1–2% of these patients become symptomatic.Although duodenal diverticula constitute a rare cause of acute abdomen, careful imaging analysis can aid to the identification of this uncommon factor of abdominal symptomatology. Contrast-enhanced CT is the imaging of choice for a rapid, non-invasive, and specific evaluation of PAD associated with Lemmel syndrome. Oral contrast could help in confirming size-related symptoms.