A solitary fibrous tumor (SFT) is a rare mesenchymal tumor that occurs mostly in pleural sites, and an SFT occurring in the ischiorectal fossa is extremely rare. Because of the rarity, there are few reports detailing an SFT in the ischiorectal fossa.A pararectal tumor was incidentally found in a 42-year-old man during a routine medical examination. The patient had no symptoms and no previous medical history. In the physical examination, a smooth-margined and hard elastic mass was felt, and in a digital rectal examination, the rectal mucosa appeared normal. A computed tomography (CT) scan showed a 5-cm, well-defined, solid mass in the left ischiorectal fossa. Contrast-enhanced CT in the early phase showed intense heterogeneous enhancement that persisted during the delayed phase. T2-weighted images of magnetic resonance imaging yielded heterogeneous intermediate and low signal intensity. Intense arterial enhancement suggested a hypervascular nature, and persistent delayed enhancement and low signal bands on T2-weighted images suggested a fibrous component of the mass. An SFT was suspected. Most SFTs are benign but have malignant potential. Our patient did not hope for surgery if the tumor was benign; therefore, an ultrasound-guided transperineal core needle biopsy was performed to decide on a treatment strategy. Microscopic examination showed tumor cells appearing as spindle and fibroblast-like cells within a collagenous stroma. Immunohistochemistry identified CD34 and vimentin, supporting the diagnosis of an SFT. The patient consented to excision of the mass. He was placed in a prone jackknife position, and the tumor was removed transperineally using a posterior approach (modified Kraske procedure). The levator ani muscle, external sphincter muscles, and rectum were not involved and separated from the tumor. The tumor was successfully resected en bloc with no complications. Five uneventful days post surgery, the patient was discharged. There was no local recurrence during the year following surgery.Imaging findings reflect the tissue characterization such as hypervascularity and fibrous nature of SFTs. We have presented a rare case of an SFT in the ischiorectal fossa with useful imaging findings for diagnosis, treatment strategy, and successful surgical removal using a posterior approach. A solitary fibrous tumor (SFT) is a rare type of mesenchymal tumor that was initially reported to occur in the pleura. SFTs are now known to occur throughout the body . HoweverA 42-year-old man presented to our outpatient clinic because of the presence of a pararectal tumor that was found incidentally during a routine medical examination. The patient had no urinary or gastrointestinal symptoms and no previous medical history. In the physical examination, a smooth-margined, hard elastic mass was felt, and in a digital rectal examination, the rectal mucosa appeared to be normal.The computed tomography (CT) scan showed a 5-cm, well-defined, solid mass in the left ischiorectal fossa abutting the left anal wall and extending into the inferior perineum Fig. . ContrasMagnetic resonance imaging (MRI) revealed several useful findings for differential diagnosis Fig. . T1-weigAlthough contrast-enhanced CT showed nonspecific findings that were not inconsistent with a benign tumor, an SFT was suspected particularly from the MRI findings. Our patient did not hope for surgery if the tumor was benign; however, most SFTs are benign but have malignant potential; therefore, an ultrasound-guided, transperineal core needle biopsy was performed for a more precise diagnosis to decide on a treatment strategy.Microscopic examination showed tumor cells appearing as spindle and fibroblast-like cells within a collagenous stroma Fig. . ImmunohThe patient consented to excision of the mass. He was placed in a prone jackknife position, and the tumor was removed transperineally using a posterior approach (modified Kraske procedure). A 5-cm vertical incision was used to approach the mass Fig. . FollowiGross examination of the tumor demonstrated a 53 × 35 × 25-mm, well-defined, completely encapsulated mass Fig. . The secThere was no local recurrence of the tumor during the year following surgery.An SFT is a rare neoplasm that occurs in approximately 2.8 of every 100,000 people. Recent advances in electron microscopy and immunohistochemistry have demonstrated a mesenchymal rather than a mesothelial origin of SFTs. While the majority of SFTs occur within the pleura, approximately 30% of SFTs arise in the peritoneal cavity, retroperitoneal soft tissue, and pelvis [Pelvic SFTs usually occur during the fifth decade of life  and do not exhibit any gender predilection . Most paTumors involving the ischiorectal fossa are most often secondary to direct extensions of primary anorectal, prostate, and sacral tumors . PrimaryTypical SFTs show an architecture characterized by a combination of alternating hypocellular and hypercellular areas separated by thick bands of hyalinized, somewhat keloidal collagen and branching hemangiopericytoma-like vessels. Myxoid changes, areas of fibrosis, and interstitial mast cells are commonly observed. Some SFTs may contain mature adipocytes and/or giant multinucleated stromal cells, thus overlapping morphologically with the so-called lipomatous hemangiopericytomas and giant cell angiofibromas. Malignant SFTs are usually hypercellular lesions showing at least focally moderate to marked cytological atypia, tumor necrosis, numerous mitoses (≥ 4 mitoses per ten HPF) and/or infiltrative margins . Tumor cMRI shows intermediate signal intensity on T1-weighted images and heterogeneous high and low signals of mixed intensity with multiple flow voids representing prominent vascular channels on T2-weighted images , 5. On TThe differential diagnosis of primary tumors in the ischiorectal fossa with hypervascular and fibrous nature are GIST, leiomyoma, and soft tissue sarcoma. MRI findings based on tissue characterization of tumors can narrow the differential diagnosis; however, they often overlap. Therefore, further accurate and less invasive investigations for diagnosis are necessary to determine an appropriate treatment strategy.Given the perceived potential for tumor seeding along the needle tract even for SFTs , 11, perComplete surgical excision is the only established curative treatment for an SFT. However, surgery may become difficult due to extensive adhesions to adjacent vital structures. If complete surgical excision is not possible with an acceptable safety margin, local recurrence is a possibility. Generally, the posterior approaches in the prone jackknife position (modified Kraske procedure) are commonly used for incision of the ischiorectal fossa and presacral tumors , 13–16. Imaging findings reflect the tissue characterization such as hypervascularity and fibrous nature of SFTs and narrow the differential diagnosis. We have presented a rare case of an SFT in the ischiorectal fossa with useful imaging findings for diagnosis, treatment strategy, and successful removal using a posterior approach.


Yolk sac tumor (YST), which most frequently arises in the gonads as a type of germ cell tumor, is rare in children but is highly malignant. It has been suggested that alpha-fetoprotein (AFP) can be applied as a feasible tumor marker because its level was elevated in >90% of YST. The treatment generally involves debulking surgery of tumors followed by systemic chemotherapy. Metastasis process of YST in children is different from that in adults and thus the treatment option is required. In this study, we described a rare case of YST in terms of the clinical manifestation, imaging, and histopathology findings, diagnosis and treatment in an 8-year-old girl. Furthermore, it is important to investigate more thoroughly a patient with history of intermittent abdominal pain and fever with previously multiple accesses, because these might be the critical signs for YST that should be alarmed for early treatment. Although YST is rare in children, pediatric physicians should be aware of this and prompt treatment should be addressed. Ovarian germ cell tumors (OGCTs) in which neoplasms form in the germ cells of the ovary, account for about 15~20% of all ovarian neoplasms  is rare per se was grossly not involved by tumor, metastasis occurred as tumor invasion to the left periadnexal tissues and the left infundibulopelvic ligament were noted, thus left salpingo-oophorectomy was also done in order to prevent residual tumors and possible secondary surgery.An 8-year-old girl suffered from intermittent abdominal pain and fever for 3 weeks. She had visited local clinics and regional hospital for several times. Oral medication was prescribed for pain and constipation, but her symptoms persisted. Then, she was brought to the Pediatrics outpatient department with nausea, vomiting and lower abdominal pain for 2 months. On physical examination, lower abdomen tenderness, and pale looking were noted. The ultrasonography (US) showed a huge pelvic tumor measuring 11.1 × 9.4 cm in size with cystic mass (3.6 × 2.8 cm) and bloody ascites over right subhepatic and lower abdominal area. There was no family history of cancer. The clinical diagnosis was pelvic mass, possibly neoplastic in nature. Computed tomography (CT) revealed a large pelvic mass, measuring 12 × 9.7 × 7.38 cm in size with internal heterogeneity and gynecological origin (ovary or urterus), enlarged paraaortic and mesenteric lymph nodes and fluid in Morrison pouch . A threeThe patient was discharged 5 days after operation and recovered well. Gross examination of the specimen submitted for pathology showed a piece of necrotic tissue, measuring 3.5 × 2.0 × 0.3 cm in size. H&E stained sections of the specimen revealed tumor necrosis with small focus of atypical cells with hyperchromatic nuclei mainly in glandular or reticular pattern. Immunohistochemical staining revealed the residual cells were immunoreactive to AFP and glypican-3 (GPC-3). The findings were consistent with YST. The patient was then referred to the oncologist for further treatment and under stringent follow-up.YST is a rare tumor of childhood, which account for ~3.5% of all childhood cancers (<15 years) and usually arises in gonads, testis or ovary, thus a type of germ cell tumor. The incidence between the ages 15 and 19 rises to 16% . It has There are a number of diagnostic tools applied for YST, such as US, CT, magnetic resonance imaging (MRI) and histopathological analysis . US charIt has been suggested that AFP can be applied as a feasible tumor marker because its level was elevated in >90% of YST . In our The exact pathogenesis of YST remains obscure. However, some studies propose that it occurs from malignant transformation of misplaced germ cells. During the 4–6th week of embryogenesis, germ cells migrate laterally between the embryonic ectoderm and endoderm; germ cell tumor can arise anywhere along the migration from the mesoderm to the future cranial area . TherefoThe general treatment for YST is surgery for eliminating the primary tumor without severe morbidity  Table 1Table 1. Our case highlights the importance of YST in children, and we provided our valuable experiences in the approaches of diagnosis and treatment for YST in children. Furthermore, it is important to investigate more thoroughly a patient with history of intermittent abdominal pain and fever with previously multiple accesses, because these might be the critical signs for YST that should not be neglected in order to treat the patient earlier. Although YST is rare in children, pediatric physicians should be still aware of this as it can be fatal and prompt treatment should be addressed.We have obtained verbal and written informed consent from the patient's guardian for the publication of this case report.LC, K-CY, and S-BY wrote the manuscript. H-JW reviewed and revised the manuscript. All authors approved the final version of this manuscript.The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.


Esophageal perforation is a rare presenting sign of gastric cancer. To date, only nine case reports of this phenomenon have been previously published.Esophageal perforation was diagnosed radiographically during workup for acute chest pain in a 67-year-old man. Emergent endoscopy confirmed esophageal perforation and biopsied a pre-pyloric mass confirmed to be adenocarcinoma. The perforation was managed with endoscopically placed transluminal pleural and mediastinal drains and esophageal stenting. The gastric outlet obstruction was temporized with a transpyloric stent. After the patient recovered from sepsis, distal gastrectomy was performed and he made a full recovery.Rarely, pre-pyloric gastric cancer can present with Boerhaave syndrome, spontaneous esophageal perforation associated with forceful vomiting. We present the tenth report in the literature of this phenomenon and the first to be initially treated with endoscopic stenting and transluminal thoracoscopic drainage. When endoscopic management is used to treat patients with Boerhaave syndrome, it may be beneficial to examine the entire stomach to evaluate for malignant etiology. Gastric cancer is a major source of global burden of disease and mortality. Though it is the fifth most common malignancy worldwide, it is the third leading cause of cancer deaths . The majA 67-year-old man presented to the emergency room for acute chest pain, dyspnea, and chills. On further questioning, he reported 1 month of nausea, vomiting, and dysphagia and an unintentional 90-lb weight loss over the past year. Social history was notable for tobacco abuse of one pack per day and occasional alcohol use. Family history was significant for unspecified malignancy in both parents and a daughter, as well as gastric cancer in a brother and granddaughter. On exam, he was tachycardic (heart rate 110 beats/min), hypotensive (blood pressure 86/68 mmHg), and tachypneic (respiratory rate 40 breaths/min) and had diffuse left costal chest tenderness. Given his history of hypertension, cardiac workup was performed and was negative. Computed tomography (CT) of the chest was obtained and revealed an esophageal perforation and thickening of the gastric pylorus  showed a large perforation of the distal esophagus just proximal to the gastro-esophageal (GE) junction, a large fluid- and food-filled stomach, and a large ulcerative pre-pyloric mass , with a 1.8-cm moderately differentiated gastric adenocarcinoma with negative margins, and two of two lymph nodes evidencing metastatic disease including focal extra-nodal extension.The patient required hospitalization for nearly 3 months for infection control, nutritional support, and physical rehabilitation. Four serial endoscopies were performed during this time for esophageal evaluation  from gastric cancer and the first to be temporized by esophageal and pyloric stenting with transluminal perforation management. Previously, Matsuhashi and colleagues  publisheSignificantly, the location of the tumor in all ten cases was pre-pyloric. The pathophysiology of the spontaneous esophageal perforations observed in these patients may be explained by malignant invasion of the pylorus inciting forceful vomiting akin to pyloric stenosis. Furthermore, it is likely that the stomach was unable to diffuse much of the force of retching as it was already maximally distended from months of accumulation of fluid and particulates secondary to poor gastric outflow.Boerhaave syndrome, spontaneous esophageal rupture due to a sudden increase in intra-esophageal pressure often due to forceful vomiting, is a rare phenomenon with a mortality rate of about 40% . Treatme


A 22-year-old man has consulted in emergency for acute urinary retention and left renal colic. Bladder catheterization was performed. Symptomatic treatment was provided with no improvement. MRI showed a pseudotumoral bladder wall thickening associated with vesical floor budding with prostate median lobe infiltration. The patient got an endoscopy that concluded to an inflammatory aspect of the bladder mycosa and a solid mass in the bladder neck arising. The biopsy during examination concluded to a glandular cystitis. Ultrasonography performed six months later still showed an enlarged prostate of 60g volume, post void residue of 280ml and bilateral hydronephrosis. A second cystoscopy showed an obstructive prostate with a median lobe. A transurethral resection of this lobe was performed. The pathological examination concluded to a benign prostate hyperplasia. This case is likely to be the first reported so far about a BPH in a young male associated with Cystitis Glandularis. Neither etiology nor evidence of the cause behind this case has been identified so far. Although Benign Prostate Hypertrophia is rare among young males, its ethiopathogenesis is not well known, its relation with cystitis glanduralis in young patients has never been described before. Both medical and surgical approaches remain similar to the adults. Benign prostatic hyperplasia (BPH) is a common disease that usually develops in middle-aged men. It is very rare in the third decade of life, around 7cases have been reported so far and no cases of associated BPH and cystitis glandularis has been reported so far in young patient.A 22-year-old man, with no past medical history, has consulted in emergency for acute urinary retention and left renal colic. Bladder catheterization was performed as well as an ultrasound that showed left obstructive premeatic calculi of 6mm and bladder distension. On examination the patient was afebrile and a slight tenderness on the left flank was found on abdominal palpation. On DRE we found an enlarged prostate, there were no indurations and no areas of softness or tenderness. An anti-inflammatory and antalgic treatment was provided to the patient. After one week we take off the bladder catheterization but the patient still presents a dysuria. Another ultrasound was performed; there was an inflammatory aspect of the bladder with a thickened bladder wall and a tissular proliferation measuring 5*2 cm on the bladder neck and at the left wall with large implantation and probably prostate infiltration. It was vascularized on color Doppler ultrasound . MeanwhiThe average prostate volume from the age of 1 to 10 is 1.4cc (0.4g) and 10.8cc 3.8g) from 11 to 20. From 21 to 30, it is 19.8cc .8g from  reportedth, 2017. He did not report any complaints, nor voiding troubles or sexual ones so far.However, BPH was associated to cystitis glandularis with an unusual proliferative disorder of the urinary bladder, which is characterized by transitional cells that have undergone glandular metaplasia . Its potAlthough benign prostate hypertrophia is rare among young males, its ethiopathogenesis is not well known. Some hypothesis have been studied as causing BPH, but their relation with cystitis glanduralis in young patients has not been described before, and the cause remain unknown. There is no specific treatment for young males for BPH and for Cystitis, and both medical and surgical approaches remain similar for adults.The authors declare no competing interests.


An enterocele is a pelvic hernia formed from the separation of endopelvic fascia, associated with the posterior or anterior vaginal fornix, and most commonly located in the posterior superior vaginal segment. Rectal prolapse is a debilitating condition in which the mucosa of the rectum protrudes circumferentially from the anus. Surgical repair is the recommended treatment for rectal prolapse, and though there are many different surgical options, there is no consensus on which approach is best. We present a case of anterior rectal prolapse due to enterocele which was treated by correction of enterocele with a vaginal approach and propose some clinical features and diagnostic techniques that may distinguish this entity from traditional rectal prolapse. An enterocele is a pelvic hernia formed from the separation of endopelvic fascia, associated with the posterior or anterior vaginal fornix, and most commonly located in the posterior superior vaginal segment . As a trAn 84-year-old postmenopausal white female  was referred to our urogynecology clinic for evaluation and treatment of a persistent rectal bulge  . She Pelvic exam revealed an atrophic vagina with no significant anterior or posterior vaginal wall prolapse. Her cervix was absent surgically, and her pelvic organ prolapse quantification was as follows: GH 2, PB 2, Aa -3, Ba -3, Ap -3, Bp -3, C -7, D n/a, and TVL 7. Rectal exam showed normal perianal skin and sphincter tone. With Valsalva, she demonstrated significant anterior rectal wall prolapse with palpable contents consistent with small bowel enterocele in the rectovaginal space. Prolapse was approximately 6 cm in size with the anterior rectal wall extending outside the anal verge and no evidence of posterior or circumferential rectal prolapse. Endoanal ultrasound showed normal levators, intact external anal sphincter circumferentially, and attenuation of the internal anal sphincter. There were no discernible masses, but the patient was unable to produce her rectal prolapse with the ultrasound transducer in place. Transperineal ultrasound was used to dynamically evaluate the perineal and rectal anatomy and demonstrated a large enterocele prolapsing between the vagina and the rectum with apparent fat or small bowel contents. Prolapse at the anterior rectal wall was directly associated with this enterocele. Defecography and dynamic MRI were not available at our facility.The patient was evaluated by a colorectal surgeon for opinion on treatment plans. This surgeon indicated that a rectopexy would be acceptable, but it would be reasonable to attempt to repair the enterocele first to see if her symptoms would resolve and to potentially spare the patient from a more morbid procedure. The patient indicated a desire to proceed with an attempt at repair with the vaginal approach.The patient was admitted to the hospital and anesthetized for surgery. A vertical posterior vaginal incision was made, extending into the perineal body. The vaginal epithelium was separated from the underlying endopelvic fascia, and dissection was performed superiorly and laterally to the level of the ischial spines and levator ani. A large enterocele was identified, opened, ligated with two purse-string sutures of delayed absorbable monofilament suture, and then excised Figures  and 3. APostoperatively, the patient recovered well and was discharged home on the first postoperative day with complete resolution of her rectal prolapse symptoms. She was seen at 1 month and 3 months postoperatively and by 3 months had resumed her normal activities of daily living with complete symptom resolution and normal defecation without incontinence.Rectal prolapse is a challenging and debilitating disease, with significant negative impact on sufferers. A significant number of surgical procedures (>100) are available to apply in these cases, and there is no consensus on which procedure is best . CurrentAs noted in Devakumar's discussion, Moschowitz proposed in 1912 that rectal prolapse could be the result of a deep pouch of Douglas which would allow pressure on the anterior rectal wall to cause the rectum to evert outside the anus , 8, and It is important to note that there are likely significant differences between these two cases and traditional circumferential rectal prolapse. It is possible that these are unique clinical entities. In looking at the images in Devakumar's case and in our case  , it cEnterocele presenting as anterior rectal prolapse appears to be a rare condition, but one that may be amenable to a vaginal approach to treatment rather than traditional rectal prolapse repair. Careful physical examination is crucial. As a key physical exam finding, absence of circumferential mucosal folds and isolated anterior prolapse may herald the presence of an enterocele and should prompt an appropriate evaluation. There are no studies or case series describing the ideal approach for surgical correction of this rare presentation, and as in the case of any rectal prolapse, careful individualized evaluation and planning are warranted. Further research into the mechanism of rectal prolapse and the role of enterocele is needed. In this case, a vaginal surgical approach resulted in excellent results, at least in the short term, and allowed the avoidance of potentially more invasive and morbid procedures.


Vascular leiomyomas (VL) are benign lesions of perivascular origin. We report two new cases and discuss their clinical, histological and immunohistochemical characteristics, in order to facilitate the diagnosis and treatment of such lesions. The patients, both male, presented asymptomatic nodules located in the bottom of the labial sulcus and buccal mucosa. In the second case, color doppler ultrasonography was performed, which showed no change in blood flow. After excisional biopsies, a limited lesion was observed histologically, with multiple tortuous vessels of varying sizes and calibers, and among them, spindle cells bundles, positive for smooth muscle actin. Oral VLs have clinical features similar to those of other more common lesions, making preoperative diagnosis difficult. Imaging examination, such as color doppler ultrasonography, may help in diagnosis. In general, excisional biopsy is performed, due to the ease of removal of the lesion during surgery. The treatment of choice is the complete excision of the lesion, which has an excellent prognosis and a low rate of recurrence. Key words:Vascular leiomyoma, spindle cells, thrombus, Doppler. Leiomyomas are benign neoplasms originating from smooth muscle, which occur, more commonly, in the female genital tract and rarely in the oral cavity . ClinicaThe aim of this study is to report two uncommon cases of oral vascular leiomyoma and to discuss their clinical, histological and immunohistochemical characteristics, in order to facilitate the diagnosis and treatment of such lesions.Case 01 – A 60-year old male patient presented to the dentist complaining of “swelling of the gums” in the upper anterior region, with 4 months of evolution. The extraoral examination revealed an effacement of the nasolabial fold and lifting of right nasal alar region. On intraoral examination, an asymptomatic 1cm submucosal nodule was observed filling the anterior maxillary labial fold, lateral to the midline. The lesion presented a rubbery consistency on palpation and similar coloration to the adjacent mucosa  shows images with well-defined margins and homogeneous structure, which may indicate the benign nature of the lesion. The Doppler US usually suggests the presence of intratumoral vascularity . In caseMicroscopic diagnosis may also be difficult due to features common to other spindle cell lesions, such as neurofibroma, schwannoma or fibroma. In these cases, staining such as Masson’s trichrome and immunohistochemical reactions should be considered to investigate the origin of the lesions, and the positivity for smooth muscle actin confirms the smooth muscle origin of neoplastic cells. The malignant counterpart of leiomyoma, leiomyosarcoma, should be evaluated and ruled out. The morphological difference between them is the degree of cellular pleomorphism, nuclear atypia, hyperchromatism, necrosis and number of mitoses, which are the main aspects to be taken into account .The abundant presence of mast cells in some areas of leiomyoma from the extra-peritoneal cavity in the lower abdomen, which appear to be related to angiogenesis and tumor growth, was reported previously . AlthougSmall traumas, venous stasis or hormonal causes have been suggested as the etiology of this lesion, however, it remains unknown ,10. In sThe treatment of choice for VL is complete surgical excision, with an excellent prognosis and few recurrences have been reported . DespiteIn conclusion, oral VL have clinical features similar to those of other common lesions, making preoperative diagnosis difficult. Imaging examination, such as color Doppler ultrasonography, may help in diagnosis. In general, excisional biopsy is performed, due to the ease of removal of the lesion during surgery, and although it has vascular components, excessive bleeding during the procedure is not common. The treatment of choice is the complete excision of the lesion, which has an excellent prognosis and a low rate of relapse.


Amyloidosis is the deposition of an extracellular fibrillar protein in the tissues leading to organ dysfunction. Laryngeal amyloidosis is a rare phenomenon. We report a case of isolated laryngeal amyloidosis which was initially suspicious for laryngeal cancer on magnetic resonance imaging (MRI) but histopathology showed the presence of amyloid. Systemic workup was negative. The patient is being managed conservatively. Extracellular deposition of an abnormal fibrillar protein in the tissues is termed amyloidosis . The incAmyloidosis most commonly affects men around the age of 50 to 70 years . TreatmeIn the head and neck, the larynx is one of the frequent sites of amyloid deposition . It may A 31-year-old male presented with stable dysphonia for four years. There were no sinonasal complaints, dyspnea, dysphagia, sore throat or a cough. Direct laryngoscopy exam showed left supraglottic/false vocal fold submucosal fullness extending to the aryepiglottic fold with papillomatous changes to the mucosa of the false vocal fold  of the neck revealed an ill-defined enhancing soft tissue mass centered in the left false vocal cord and paraglottic fat, extending to the left true vocal cord, anterior commissure, and left aryepiglottic fold Figure . ImagingTwo possible mechanisms have been hypothesized leading to the development of isolated localized amyloidosis . The firThe most common presentation of laryngeal amyloidosis is hoarseness, even though a cough, hemoptysis, dysphagia or dyspnea may be present -4. A revLaryngeal amyloidosis is most often treated surgically ; howeverNew therapies such as anti-amyloid antibodies are under trial for systemic disease. These have the ability to dissolve amyloid deposits  thus redAmyloidosis of the larynx is a rare disease, often presenting with hoarseness. Clinical suspicion for this entity should be high in young patients with slow-growing laryngeal mass. Treatment currently consists of surgical resection though newer therapies for amyloidosis may lead to better outcomes.


We herein reported two rare surgical cases of primary combined hepatocellular-cholangiocellular carcinoma, intermediate-cell subtype (CHC-INT), showing potentially characteristic and specific gross findings on their cut surface: both CHC-INTs demonstrated poorly demarcated and expansive and/or infiltrative hepatic nodules in lobulated margins, appearing clearly whitish in color. We were finally able to accurately diagnose the current lesions after thorough analyses including an appropriate and wide panel of immunohistochemical antibodies. Despite that, all pathologists should be aware that the potentially characteristic gross features of primary CHC-INT might also be one of the powerful supplementary tools for reaching its correct, conclusive diagnosis. In 1903, Wells first reported the features of primary malignant liver tumor, combined hepatocellular- (HCC-) cholangiocellular (CCC) carcinoma (CHC), and following that, Allen and Lisa, Goodman et al., and Taguchi et al. subclassified CHC variously . FurtherThe first patient, who was a man in his late seventies with an unremarkable previous medical history, presented with isovascular nodule accompanied by slow venous wash-out on abdominal dynamic CT in the left lobe of liver. The laboratory data, including the blood cell count, chemistry, and tumor marker levels, were within the normal limits, with the exception of mildly elevated CRP 0.39 mg/dL) and decreased hemoglobin (8.9 g/dL) levels. Neither infection of HBV nor infection of HCV was noted. Based on the clinical findings, the initial diagnosis by the clinicians was most likely HCC, and, thus, left partial hepatectomy was performed. On gross examination, the cut surface of hepatic nodule  showed a9 mg/dL aThe second patient, who was also a man in his early seventies with more than 25-year follow-up for HCV-positive chronic hepatitis and recurrent HCC, presented with mildly hypervascular and ring-enhanced nodule accompanied by venous wash-out on abdominal dynamic CT in the S5 of the remnant liver. The laboratory data were mostly within the normal limits, with the exception of mildly elevated AST 37 IU/L), total bilirubin (1.8 mg/dL), and CEA (5.55 ng/mL) levels. No infection of HBV was observed. Based on the clinical findings, the initial diagnosis by the clinicians was recurrent HCC, and, thus, partial S5 hepatectomy was performed. On gross examination, the cut surface of hepatic nodule  showed a7 IU/L, tIt is very likely that the current report of two surgical CHC-INT patients is clinicopathologically remarkable for one reason at least. In case of the present gross findings for poorly demarcated, unencapsulated, and lobulated nodules looking clearly whitish in color on the cut surface, we pathologists should consider the rare possibility of CHC-INT, rather than common HCC. In addition, a wide panel of immunohistochemical analyses should be critically performed, as shown here. In our opinion, the macroscopic features of HCCs appear yellow-whitish, tan to green, but not clearly whitish, in color, even though further modified by varying degrees of necrosis/hemorrhage and the production of bile or fat. In fact, the most critical differential diagnosis in the present cases was HCC, despite the fact that it should be relatively easy to rule out this possibility through a clinicopathologic examination or immunohistochemistry. Since most CHCs are known to generally have a worse outcome, growing rapidly and showing aggressive/infiltrative behaviours , 4, 6, aIn conclusion, we herein reported two rare surgical cases of primary CHC-INT, showing potentially characteristic and specific gross findings on their cut surface: both CHC-INTs demonstrated poorly demarcated and expansive and/or infiltrative hepatic nodules in lobulated margins, appearing clearly whitish in color. We were finally able to accurately diagnose the current lesions after thorough analyses including an appropriate and wide panel of immunohistochemical antibodies. Despite that, all pathologists should be aware that the potentially characteristic gross features of primary CHC-INT might also be one of the powerful supplementary tools for reaching its correct, conclusive diagnosis.


We present a case of spontaneous undifferentiated/unclassified sarcoma, of a pleomorphic subtype formerly known as malignant fibrous histiocytoma (UPS/MFH), arising from the pancreas of a laboratory rat. The mass was excised after laparotomy from a 6‐month‐old female laboratory Wistar rat. It presented a giant multilobulated mass of irregular shape, which had arisen from the pancreas and occupied almost the entire peritoneal cavity. Histologically the tumor was characterized by a highly variable morphological pattern, with frequent transitions from storiform to pleomorphic areas. An extensive immunohistochemical examination revealed no specific lines of differentiation. Immunohistochemical positivity was observed only to MIB‐1 (high Ki‐67 proliferation index), vimentin and CD68 antibodies. The diagnosis was compatible with UPS/MFH. To the best of our knowledge, the present case is the first report of a spontaneous primary UPS/MFH arising from the pancreas of a laboratory rat. It was group‐housed in a Makrolon cage, with two other rats, at 20‐22°C room temperature, on a 12‐hour light/12‐hour dark cycle and was provided with commercial pelleted diet and tap water ad libitum. The facilities and experiments were in accordance with Directive 86/609/EEC for the care and use of laboratory animals.After midline laparotomy, a tumor arising from the pancreas was revealed and excised. This was a solid encapsulated multilobular mass of irregular shape Figure . No tumoMacroscopically, the excised mass weighed 26 g, was 10.1 cm in length and 3.5 cm at its maximum width. The cut surface of the mass was solid, of a yellowish‐white color with dark reddish and white areas  stained sections. In addition, two‐step immunohistochemical staining was performed using the biotin complex EnVision + system (Dako Cytomation). Antibodies against the following antigens were used: vimentin, CD‐68, desmin, SMA, S100 protein, EMA, cytokeratin AE1/AE3, CD117, MIB‐1(Ki‐67), and CD34 (Dako Corporation). Finally, bound antibody complexes were stained for 10 minutes with 0.05% diaminobenzidine. The histochemical and immunohistochemical stained sections were examined under a Nikon Eclipse 50i microscope.3Histopathological examination revealed a highly variable morphological pattern with frequent transitions from storiform to pleomorphic areas and scattered areas of necrosis. The lesions mainly consisted of plump spindle cells arranged in short fascicles in a cartwheel or storiform pattern around slit‐like vessels and pleomorphic areas with plump fibroblastic‐like cells and rounded histiocytic‐like cells arranged haphazardly, without any particular orientation with regard to vessels Figure A. PleomoAccording to the current WHO classification, the diagnosis was compatible with a high‐grade undifferentiated/unclassified sarcoma of pleomorphic subtype.4Primary UPS/MFHs are rarely localized to the pancreas, with less than 25 cases of previously termed MFHs or UPS/MFHs being reported in humans to date.The only spontaneous pancreatic sarcomas recorded to date in the rat have been a hemangiosarcomaIn the present case, a UPS/MFH tumour, arising from the pancreas and occupying almost the entire peritoneal cavity, was revealed in a 6‐month‐old female rat. This was an exceptionally large mass (10.1 cm in length) compared to human pancreatic UPS/MFHs, whose largest recorded dimension is 35 cm.In our case study, immunohistochemical analysis revealed no specific lines of differentiation. Immunohistochemical positivity for CD68  is frequently reported in undifferentiated sarcomas, and this was observed in the present case. It therefore seems that this demonstrates the existence of high numbers of tumor infiltrating histiocytes rather than true histiocytic differentiation of the neoplastic cells.The size of the tumor and the depth of its location are considered the two most important prognostic factors in evaluating the capacity to metastasize of tumors formerly known as MFHs.5The present case is the first report of a spontaneous primary undifferentiated/unclassified soft tissue sarcoma of the pleomorphic subtype arising from the pancreas of a laboratory rat. The tumor was an exceptionally large pancreatic UPS/MFH. Based on the age of the rat, we can assume that this type of malignancy may develop in early adulthood or even in childhood.None.PY performed the laparotomy and mass excision, carried out data management, participated in designing the study, researching the background literature and drafting of the manuscript. ML, SM, and TP performed histopathology, histochemical and immunohistochemical examinations, evaluated the results, researched the background literature and drafted the manuscript. CS participated in the coordination and drafting of the manuscript. All the authors read and approved the final manuscript.


The dignity of adrenal masses in children varies from benign lesions like adenoma and ganglioneuroma to malignant tumors like adrenocortical carcinoma and neuroblastoma. Any surgical approach, especially minimally invasive surgery (MIS), requires careful risk stratification based on oncological and technical criteria. Herein, we present four patients who underwent MIS for adrenal masses. Laboratory testing differentiated between simple cysts and adenoma, but could not identify a child with adrenocortical tumor preoperatively. Analysis of image-defined risk factors excluded vascular encasement in all cases. All patients underwent laparoscopic adrenalectomy without complications. Histopathology revealed simple cyst, ganglioneuroblastoma, adenoma, and potentially malignant adrenocortical tumor in one patient/case each. All specimen showed clear margins and no recurrence was noted at a mean follow-up of 18 months. The adrenal gland consists of a medulla and a cortex and may give rise to various types of tumors. While adenomas and carcinomas may develop in the cortex, the medulla may be the origin of neuroblastomas, pheochromocytomas, and ganglioneuromas. All of these lesions may require surgical intervention by an open or a minimally invasive approach. The risks and benefits of pediatric laparoscopic adrenalectomy for malignant or semibenign lesions remain a matter of vivid discussion. While simple adrenal cysts or adenomas are benign lesions and a minimally invasive approach may be considered, adrenocortical carcinomas and (ganglio)neuroblastomas require careful assessment of risk factors such as serum biomarkers, hormonal status, and image-defined risk factors (IDRFs) .A 3-year-old boy presented with precocious puberty (tanner stages P2 and G2). X-rays of the extremities showed an accelerated bone age of ∼6 years. Ultrasound (US) and magnetic resonance imaging (MRI) of the abdomen revealed a right adrenal mass of approximately 4.0 × 2.5 × 4.0 cm . The laA one- and half-year-old female presented with a precocious puberty (virilization with clitoris hypertrophy and pubarche P2–3). Laboratory parameters are shown inA 15-year-old male patient presented with exercise-triggered recurrent right abdominal pain. US and MRI demonstrated an uncomplicated cystic structure in the right adrenal gland with sharp delineation to the upper renal pole and without pathological contrast enhancement  giving A 7-year-old boy presented due to recurrent abdominal pain. The routine US revealed an inhomogeneous expansion of approximately 3 × 3 × 2 cm with calcifications in the right adrenal gland. Laboratory data are displayed inIn this case series, we present four pediatric patients with a broad spectrum of the adrenal gland masses undergoing laparoscopic resection.2insufflationGenerally, tumors of the adrenal gland can be malignant  or benign .Guidelines for adrenalectomy poorly define the indications for MIS in the pediatric age group. The rarity of adrenal lesions in this age group, a wide pathologic spectrum, the predominance of malignant lesions, large dimensions of the masses in relation to small body size of the patients, and technical challenges with miniature instruments may be hypothesized as the underlying reasons.After initial suspicion, patients with adrenal masses undergo routine diagnostic workups usually involve imaging  paired with laboratory analysis . Concerning imaging special emphasis is put on the presence or absence of IDRFs. Infiltration of the renal pedicle, encasement of major vessels , the porta hepatis, dumbbell tumors, muscular infiltration, and compression of kidney or ureter have been identified as IDRFs.While MIS is the mainstay in benign lesions,Special care has to be taken to identify the rare case of a malignant ACT. Suspicion should be raised in case of adrenal masses presenting with IDRFs and especially in patients with signs of precocious puberty. ACTs have a high risk of intraoperative rupture (∼20% of cases) with consecutive tumor spillage.Taken the recent literature into account, the indication for MIS was clear in cases 1, 3, and 4 presented in this series. Histology of case 2 revealed a potentially malignant adrenocortical tumor raising the question, if MIS was the optimal therapy for this patient. As there was no evidence of IDRFs and the tumor size was < 5 cm, MIS was performed. Although an open procedure may have been beneficial, the tumor could be removed completely by MIS and the patient is free of recurrences until now .In conclusion, MIS is indicated in benign lesions, but has to be discussed critically in case of potential malignancy. In these cases, a MIS approach may be considered in selected patients without IDRFs as long as the principles of oncologic surgery can be maintained.


Laparoscopic adjustable gastric banding is commonly used to treat obesity. It rarely results in complications, one of which is gastrointestinal erosion. Simultaneous erosion of the stomach and colon is a rare finding that has been documented in only a few case reports. We present a 62-year-old female with abdominal pain, nausea, and hematochezia. She was found to have simultaneous gastric and colonic erosion identified on CT scan. Imaging findings were confirmed and device was removed during surgery. Laparoscopic adjustable gastric banding (LAGB) uses a restrictive mechanism to reduce the stomach’s capacity and subsequently limit caloric intake in obese patients. In 2015, LAGB comprised 5.7% of all bariatric surgery cases.2 LAGB has several advantages including low mortality rate, adjustability of the outlet to allow flexibility with caloric needs, reversibility, less surgical complications and avoidance of staple lines and prosthetic mesh used in other bariatric techniques. However, LAGB is rarely accompanied by complications that may lead to serious consequences for the patient. In this case, we will discuss a rare complication: simultaneous gastric and colonic erosion after LAGB. To our knowledge, only three similar cases have been previously described in the literature.According to the Centers for Disease Control and Prevention, over one-third of Americans are obese.A 62-year-old lady with past medical history of lupus anticoagulant on warfarin, and grade II obesity (body mass index of 38.5) status post laparoscopic gastric banding in 2003, presented with a 3-day history of bright red blood with clots per rectum. She presented with left-lower quadrant abdominal pain, nausea, emesis, lightheadedness, weakness and exertional dyspnea. On evaluation in the emergency department, patient was afebrile and vital signs were within normal limits. Physical examination was unremarkable. Haemoglobin was 9.4 and INR was 2.1 on admission.Owing to bleeding per rectum, colonoscopy was performed which showed diverticulosis and a foreign body eroding through the transverse colon. CT scan of the abdomen with oral contrast was ordered by the referring physician. It should be noted that the use of positive oral contrast agents is not recommended because it can obscure mucosal enhancement and intraluminal haemorrhage. Ideally, the study should be ordered with and without IV contrast medium. The CT scan showed erosion of the lateral portion of the laparoscopic gastric band into the wall of the stomach . There wThe leading diagnosis after reviewing the CT scan of abdomen was gastric and colonic erosion secondary to laparoscopic gastric band. However, other differential diagnoses included diverticulitis, gastroenteritis and gastrointestinal perforation.Bariatric surgery was consulted and patient was taken to the operating room for laparoscopic removal of eroded gastric band, repair of gastric erosion and repair of transverse colon erosion.Approximately 5 months after surgery, patient was seen in clinic and was doing well from a GI standpoint. She had lost 8 pounds since the surgery.3 The connecting tube may also cause erosion of the distal colon. When gastric band erosion is identified on CT scan, it is important to trace the length of the connecting tube for possible involvement of the distal GI tract, as simultaneous erosion of the colon has been documented.5 In two previously documented cases, gastrocolic fistulas were formed.5 One of these patients also had an associated abscess formation.5 Yet another case reported evidence of small bowel obstruction secondary to connection tube.6 Our case is unique in that there was no evidence of associated fistula formation, abscess or small bowel obstruction.Gastric band erosion is an important late complication with an overall incidence of 1.46% of cases as reported in a recent systematic review.8 Clinical presentation of gastrointestinal erosion may include nausea, emesis, abdominal pain and hematochezia, or patient may be asymptomatic. Of all cases with simultaneous gastric and colonic erosion, our patient was the only one to report haematochezia as the presenting symptom. This may be related to the patient’s long-term use of anticoagulants. Endoscopy and/or colonoscopy may show foreign body as seen in our case. Furthermore, CT scan of the abdomen may show displaced gastric band inferior to gastric fundus, erosion of band into the gastric wall, erosion of connecting tube into colon with surrounding inflammatory changes, gastrocolic fistula or abscess. In previous documented cases, the complete diagnosis of gastric and colonic erosion was confirmed with the addition of gastroscopy or during surgery in addition to CT findings. In contrast, CT scan alone was sufficient to confirm both gastric and colonic erosion in our patient. Treatment involved band removal with repair of gastric and colonic erosions using laparoscopic technique, which was similar to previous cases.Other rare complications of LAGB include stomal stenosis, malpositioned bands, pouch dilation, band slippage, perforation, gastric volvulus, intraluminal band erosion and port- and band-related issues.An important limitation of this case report includes obtaining the CT scan with oral contrast only, instead of the correct approach of with and without IV contrast medium. In patients with suspicion of GI bleeding, the use of IV contrast allows the radiologist evaluate for active bleeding and ischemic changes in the bowel walls. It also increases the sensitivity for distinction between the visceral wall and the laparoscopic gastric band device. Unfortunately, this study was ordered and performed with oral contrast only.10 Given that band erosion is a late complication that may not appear for several years after the procedure, it is important to have a low threshold for this diagnosis. We hope that this case report will help radiologists and clinicians to keep this rare complication on their differential diagnosis in future cases with similar presentations.Several mechanisms have been suggested to correlate with band erosion, including chronic ischaemia secondary to gastric wall pressure from the band, suturing the band to the stomach, infection and even number of cases performed by the bariatric surgeon.In a patient with laparoscopic gastric band presenting with haematochezia, nausea, emesis and abdominal pain, band erosion should be considered in the differential diagnosis.Once gastric band erosion is identified on CT, one should trace the length of the connecting tube in search for possible colonic erosion.Treatment for simultaneous gastric and colonic erosion involves band removal with repair of gastric and colonic erosions using laparoscopic technique.Written informed consent for the case to be published  was obtained from the patient(s) for publication of this case report, including accompanying images.


Retroperitoneal schwannomas are rare.We here report a case of 64-year-old woman who was referred to her local hospital for abdominal pain and found to have a palpable tumor. Computed tomography (CT) and colonoscopy revealed a combination of liver and colon lesions and colon cancer with a large liver metastasis was suspected. After neoadjuvant chemotherapy had proved ineffective for her presumed liver metastasis, the patient was referred to our hospital where we performed a simultaneous right hemicolectomy and extended left hepatic lobectomy. The pathological diagnoses were a colonic adenocarcinoma and retroperitoneal schwannoma immediately adjacent to the liver.Although liver metastasis should be the first provisional diagnosis in patients with advanced colon cancer, retroperitoneal schwannoma should also be suspected in the differential diagnosis of possible liver lesions. Herein, we report a case of retroperitoneal schwannoma preoperatively diagnosed as a liver metastasis from colon cancer for which the patient underwent a simultaneous left hepatic lobectomy and right hemicolectomy. We also present a review of relevant published reports.This work has been reported in line with the SCARE criteria .2A 64-year-old woman with no significant medical history was admitted to her local hospital because of right lower abdominal pain and was found to have a palpable tumor. Laboratory tests showed severe anemia (hemoglobin concentration 6.5 g/dL) and high serum concentrations of tumor markers . Other routine biochemical profile variables were within normal limits.Computed tomography (CT) revealed a 7.0 × 6.0 cm solid tumor apparently located in the left lobe of the liver a and lum3Schwannomas, which originate in Schwann cells in nerve sheaths, are slowly growing tumors that mostly occur in peripheral nerves in the skin and subcutaneous tissue of the head and neck; however, they can originate in other organs such as the gastrointestinal tract  and boneAlthough most cases are asymptomatic and detected incidentally by physical examination, abdominal symptoms such as abdominal distension and pain can occur if the tumor is large and compressing adjacent organs. Strauss et al. reported 28 cases of benign retroperitoneal schwannomas from a single center, 13 of whom were asymptomatic whereas eight patients presented with abdominal pain and seven with a palpable mass . The casOn pathological examination, schwannomas can be categorized into two subtypes: Antoni A and Antoni B. The majority are of Antoni A type, which is characterized by compact spindle cells arranged a palisading fashion, whereas a minority are of Antoni B type, which is characterized by collections of lipid-laden histiocytes and thick-walled, hyalinized blood vessels. Immunohistochemical staining is a useful tool to help to make a diagnosis of schwannoma because most cases have strong diffuse staining for S100 protein in the cell nuclei and cytoplasm ,7. In ouBecause there are no standards means of diagnosis, a preoperative diagnosis of retroperitoneal schwannoma is always difficult to make. CT scans typically reveal a low-density mass , whereas4We here present a rare case of retroperitoneal schwannomas which was difficult to diagnose preoperatively. Although liver metastasis should be the first provisional diagnosis when the patient has an advanced colon cancer, retroperitoneal schwannoma should also be suspected in the differential diagnosis of possible liver lesions.Huanlin Wang and other participating authors have no involvement as above.This case report is not research study, therefore approval was not given.The ethical approval has been exempted by our institution.Written informed consent was obtained from the patient for publication of this case report.TY, SI, TI, and NH performed the operation. TY, SI and NH determined the treatment plan. HW and TY prepared and wrote the manuscript. HW, YO made the pathological diagnosis. MM gave final approval for the version to be published. All authors have read and approved the final manuscript.The paper is a clinical report, no research involved.Huanlin Wang accept full responsibility for this work.Not commissioned, externally peer-reviewed.All authors have no conflicts of interest.


The hydrocele of the femoral hernia sac, an extremely rare occurrence, is termed femorocele. Very few authentically reported cases of femorocele are available in the literature. The present case, diagnosed as a case of infected femorocele, was managed successfully by excision of the femorocele sac and repair of the femoral hernia. To the best of the author’s knowledge, it is the first-ever reported case of infected femorocele.3) with neutrophilia. Ultrasonography of the right inguino-labial region revealed a mildly echogenic cystic swelling without any intra-abdominal communication. Exploration of the right inguinal region revealed a cystic (3 cm × 2 cm) swelling, medial to the femoral vessels, containing amber-colored fluid. The distal sac was excised, and anatomical repair of femoral canal defect was done after transfixing the neck of the femorocele sac. Fibro-fatty-collagenous tissue with mixed inflammatory cells along with a flattened mesothelial lining cell layer was found on histopathological examination. Sections from inguinal lymph node showed reactive hyperplasia. Culture of fluid from the sac revealed growth of Escherichia coli. The patient was put on anti-inflammatory drugs and antibiotics according to a sensitivity test. Patient was discharged in stable condition after 5 days. Four months after the operation, the patient is doing well, remaining asymptomatic and without any sign of recurrence.A 30-year-old lady presented with a painful 3 cm × 2 cm swelling in the right inguinal region. Though the swelling was there for 2 years, the pain and indurations started after a trivial blunt trauma over the swelling 7 days ago. The patient was febrile and mild tachycardic but had no dysuria. The oval-shaped, tense-cystic, poorly translucent, non-pulsatile, non-reducible swelling showed no cough impulse. There was also a (1.5 cm × 0.5 cm) palpable right-sided superficial inguinal lymph node. Routine blood and urine analysis reports were normal except leukocytosis (10,000/mmThe hydrocele of the femoral hernia sac is an extremely rare disease. When not infected, it presents a painless inguinal soft cystic swelling, commonly in women of fourth to sixth decade. This was diagnosed intraoperatively in all cases reported till date. Excision of the sac after transfixation of the neck and anatomical repair are the treatment of choice. In elderly patients, with larger defect, the mesh repair can be opted for. The femorocele may also get infected by uropathogens, and proper antibiotics should be used after a sensitivity test. Hydrocele, in females, is a rare condition. When found, it commonly involves the canal of Nuck . The hydTo the best of the author’s knowledge, it is the first-ever reported case of infected femorocele in the world.A 30-year-old lady, non-smoker with no previous major health problems, presented with fever 101 °F) and a painful swelling in the right inguinal region  without any abdominal mass, dysuria, or any sign of intestinal obstruction. She was significant only for the swelling which was located below the inguinal ligament and infero-lateral to the right pubic tubercle. It was oval-shaped, fixed to deeper tissue, tense-cystic, poorly trans-illuminant, non-pulsatile, and irreducible. The swelling was without expansile impulse on coughing and devoid of any bowel sound within it. She also had a single 1.5 cm × 0.5 cm palpable right-sided superficial inguinal lymph node. She neither had a history of any previous abdominal surgery, chronic cough, and coagulopathy nor had she received any treatment for this disease. She had no history of similar illness in her family also.3) with neutrophilia. Ultrasonography (USG) of the right inguino-labial region revealed a mildly echogenic cystic swelling of size 2.80 cm × 1.35 cm below the inguinal ligament and medial to the femoral vessels without any intra-abdominal communication or ascites  containing approximately 5 ml of amber-colored fluid Fig. . AnatomiEscherichia coli, which was sensitive to amikacin and imipenem.Sections from the sac revealed fibro-fatty-collagenous tissue with mixed inflammatory cells. There were dilated lymphatics along with a flattened mesothelial lining cell layer. This confirmed infected peritoneal sac (femorocele) Fig. . No granPostoperative period in hospital was uneventful. She was put on anti-inflammatory drugs and antibiotics targeting uropathogens . Oral feeding was started the next day. She became afebrile after 2 days, and early ambulation was encouraged. There was no surgical site infection. The patient was discharged in stable condition after 5 days. Four months after the operation, she is asymptomatic and has returned to her usual life without any clinical sign of recurrence. She was advised regular follow-up in our surgical outpatient department (OPD) to detect any recurrence by clinical examination and ultrasonography.The hydrocele of the femoral hernia sac had been an extremely rare disease. In 1882, Marcy first suggested that cystic dilatation of a portion of the femoral hernia sac may form a complication. Sir Astley Cooper was the first scientist to describe the hydrocele of the femoral hernia sac, in 1884. When Desault brought a candle near the femoral hernia sac, he found it has a transilluminated swelling . BabcockThe femoral canal is the medial-most compartment of the femoral sheath. The proximal opening of the femoral canal  is covered by a femoral septum which is condensed extraperitoneal fatty tissue. The canal normally contains loose areolar tissue, few lymphatics, and one deep inguinal lymph node called Cloquet’s node. The peritoneal sac may herniate through the femoral ring into the femoral canal, medial to the femoral vessels, to form femoral hernia. The presence of a potential or actual femoral hernia sac or embryonic peritoneal rest is the pre-requisite for femorocele formation. Different factors, like congenital, inflammatory, or traumatic occlusion of the communication with the peritoneal cavity, are responsible for the fluid collection in the femorocele sac like the hydrocele of the scrotum and spermatic cord in males and hydrocele of the canal of Nuck in females –4. FemorPrimary or true femorocele: Collection of fluid in the sac of femoral hernia either due to complete obliteration or omental plugging/adhesion at the narrow neck of the sac, with no evidence of ascites.Secondary: Fluid collection in the sac of femoral hernia from the peritoneal cavity associated with the presence of ascites.Lean females in fourth to sixth decades of their life  are at iCommonly, irreducible or incarcerated femoral hernia, cyst of the canal of Nuck, subcutaneous lipoma, Bartholin’s cyst, lymph node abscess, arterial or venous aneurysms, and post-traumatic hematoma may mimic femorocele. Also, rare entities like cystic lymphangioma, ganglion cyst, and neuroblastoma metastasis –10 in thInfection of the femorocele fluid was not reported earlier. Like the hydrocele fluid, femorocele fluid may also get infected from adjoining epididymitis/infected urinary tract by uropathogens. In the present case, trauma might have led to hemorrhage and later infection of the femorocele fluid and adjacent superficial inguinal lymphadenopathy with fever. Excision of the the femorocele sac with the repair of the defect and use of sensitive antibiotics had cured the patient.The hydrocele of the femoral hernia sac is an extremely rare disease. When non-infected, it presents a painless inguinal soft cystic swelling, commonly in women of fourth to sixth decade. This was diagnosed intraoperatively in all cases reported to date. Excision of the sac after the transfixation of the neck and anatomical repair are the treatment of choice. In elderly patients, with larger defect, the mesh repair can be opted for.The femorocele may also get infected by uropathogens and presents as pyrexia of unknown origin if one ignores its possibility to occur. When infected, it may also mimic incarcerated femoral hernia, inguinal abscess, or infected inguinal lymph node. Proper antibiotics should be used after a sensitivity test. Despite the rarity, surgeons dealing with femoral hernia should be aware of this clinical entity during the exploration of femoral hernia and suspected pathology involving the femoral hernia site.


Thyroglossal duct cyst of the tongue is a rare entity. Occurrence in the anterior part of the tongue is exceptional. We report in this paper a rare case of thyroglossal cyst of the anterior part of the tongue, discovered in a five-days-old newborn at delivery. Images have shown a cystic mass with homogenous liquid content. A transoral complete resection of the lesion was performed, with no postoperative complication. The histological analysis confirm the diagnosis of lingual thyroglossal duct cyst. There were no recurrence with a follow-up of eight months. Thyroglossal duct cyst (TGDC) represents the most common congenital midline mass. It results from remnant epithelial rests of the thyroid gland during it migration throughout the development from the tongue base . Its locA five-days-old newborn was referred to our department for an enormous lingual mass discovered at delivery. It was a normal 3,900 grams female, born by caesarean section performed because of failure to progress in labor. The Apgar score was 9/10 at 1 and 5 minutes of life. The ENT examination found a renitent mass of the tongue which measure 4 centimetres of large with a difficulty of suction but no respiratory signs . The ultet al.had treated six of nine patients with LTGDC with a needle puncture. They observed recurrence in two cases (3). The transoral complete resection without recurrence was reported [Lingual thyroglossal duct cyst (LTGDC) is a rare location, which occur in 0.3-3% of cases, and it is often interest the posterior third of the tongue . The litreported , 4, 5. Areported . In our Diagnosis of LTGDC in newborn is not usually evident. Image is essential in order to locate exactly the lesion, MRI when it is possible, is preferred because its sensitivity in soft tissues lesions characterization. Surgical approach depends on the location in the tongue and proximity to the hyoid bone.The authors declare no competing interests.


The final stage of the diagnostic of primary hyperaldosteronism is to identify the causes of excessive secretion of aldosterone and determination of its variants. Based on the analysis of literature data, the diagnostic value, sensitivity and specificity of the methods of radiation diagnostics for primary hyperaldosteronism were assessed: ultrasound, computed tomography, magnetic resonance imaging, photon emission tomography, magnetic resonance spectroscopy, scintigraphy with iodine radiopharmaceuticals. The causes of false-positive and false-negative evaluations of changes in adrenal glands in the application of these diagnostics have been analyzed. There are many genetic and morphological studies when searching the literature data on the principles and methods of distinguishing the nosological forms of primary hyperaldosteronism based on the results of the aldosterone level estimation in the separated blood from the central veins of both adrenal glands or segmental veins of one gland with subsequent determination of the concentration gradient. It was noted that topical diagnostics and, especially, the determination of nosological forms of primary hyperaldosteronism are complex and expensive, but their results allow choosing an appropriate treatment approach for each particular case. The main task of topical diagnosis is the visualization of the pathology of adrenal glands. It is performed using radial detection methods such as ultrasound, computed tomography, magnetic resonance imaging, positron emission tomography, scintigraphy with iodine-based radiopharmaceuticals.Ultrasound investigation is a widely recognized diagnostic tool in today’s clinical practice and the first method of visualization when searching for diagnosis , 2. The In many publications, the results of ultrasound scans for the detection of focal pathology of the adrenal glands are estimated to be reliably high, and in some cases, its sensitivity and specificity reach 100 % . It alloComputed tomography in the topical diagnosis of all types of adrenal cortex tumors was recognized as one of the most modern methods. It allows determining the size of the tumor, its density, structure, relationship with neighboring organs, and blood vessels. The addition of its intravascular enhancement makes it possible to detect hyperplasia or to determine the localization and characteristics of the tumor. In this case, the sensitivity of computed tomography is about 84.3 % .Compulsory conditions for computed-tomographic scanning are slice thickness of 2-2.5 mm and the use of vascular enhancement. Depending on the results, one can find a normal adrenal structure, unilateral macroadenoma (more than 1 cm), unilateral microadenoma (less than 1 cm), minimal unilateral or bilateral thickening of the adrenal glands, bilateral macro- or microadenomas or various combinations. Aldosteronomas are usually small in size, unlike other adrenocortical adenomas.•According to the results of the computed tomography scan, false-positive or false-negative evaluations of changes in the adrenal tissues, especially hyperplasia, which may accompany adenoma and may be considered as idiopathic bilateral hyperplasia, are possible. Similarly, since prolonged hyperplasia of the adrenal glands is associated with the formation of pseudo-nodal lesions, this radiographic picture is often considered to be an aldosterone-producing adenoma , 11. Add•Adrenocortical cancer with hyperproduction of aldosterone is usually evaluated as a tumor with a diameter greater than 4 cm. In this case, it is possible to detect signs of suspicion about the malignant nature of the tumor  . Most of•Small aldosteronomas can be considered as hyperplasia, especially in the bilateral process or multiple nodal lesions, which are not identified due to the small size. In addition, the so-called ‘obvious adenoma of adrenal glands’ may be sites of focal hyperplasia. Unilateral hormonally inactive adrenal macroadenomas are relatively typical for patients over 40, and they are no different from aldosterone-producing adenomas in computed tomography. Meanwhile, unilateral hyperplasia may show an increase in the size of the gland, or it is visualized as unchanged .•In the literature, based on the analysis of data on the use of computed tomography in the diagnosis and differential diagnosis of tumors of adrenal glands, the following tactics are defined:1. In the case when the formation of the adrenal gland is diagnosed earlier by other methods of research, computed tomography is recommended with low slice thickness and bolus tracking. An asymptomatic lesion less than 3 cm in size, with a computed tomography density lower than 10 HU, can most likely be considered benign. CT washout equal to or greater than 60% confirms that the lesion is benign.2. In the case of asymptomatic lesions that are detected by chance during a computed tomographic examination of the abdominal cavity organs and retroperitoneal space with contrast enhancement, a delayed scan at 15 minutes is performed. If a relative enhancement washout of 40% or higher is achieved after 15 minutes, the lesion is considered benign. Absolute values of density during the delayed phase were unreliable in the differential diagnosis.3. Heterogeneous lesions of more than 3 cm in size, detected during a computed tomography scan, which accumulate contrast agent, may cause suspicion of malignancy.4. In patients with an oncological medical history, if focal lesions of the adrenal glands are detected, one should take into account their possible metastatic nature. Computed-tomographic signs of such lesions will be variable, depending on the computed tomography pattern of the primary tumor.5. If the computed-tomographic sign of the lesion does not meet the criteria listed above, it should be regarded as uncertain. There are no specific recommendations for such lesions, and further tactics depend on the whole set of clinical symptoms and laboratory tests , 15.Magnetic resonance imaging (MRI) is successfully used in the diagnosis of adrenal tumors. In general, it does not have advantages over computed tomography scans –16.The main features and advantages of magnetic resonance imaging before other imaging methods include non-invasiveness, harmlessness, and the three-dimensional nature of image acquisition . The senDuring dynamic MRI of the adrenal glands, as in computed tomography scans, the pronounced accumulation of the contrast material and its slow elimination in malignant formations are observed, whereas the contrast material is rapidly eliminated in benign tumors .Magnetic resonance spectroscopy, developed recently, is one of the few methods that allows analyzing in vivo the chemical structure of the tissues of a living organism. Today it is the method of choice in the study of internal organs, including the adrenal glands. The method has a sensitivity of 91% and a specificity of 94% in determining the nature of the adrenal lesion in comparison with histopathological studies . Analysi1. If the lesion of the adrenal gland has typical magnetic resonance signs of fluid without a soft tissue component, further diagnostic studies are not appropriate.2. In asymptomatic tissue lesions having magnetic resonance signals and a structure similar to the adrenal gland tissue, a scan is performed 15 minutes after the injection of the contrast agent. The lesion is considered benign if it is characterized by relatively high washout rates of the contrast material (at 15 minutes).3. When the lesion of the adrenal gland was pre-visualized by other methods, a scan with a minimum thickness in different planes is recommended for differential diagnosis. In the presence of a heterogeneous magnetic resonance structure of the formation, studies using contrast enhancement are conducted. If there is a pronounced washout of the contrast agent, then we can assume that the tumor is benign.4. A large (more than 5 cm) heterogeneous lesion, which accumulates the contrast agent well and for a long time, is suspected of malignancy and requires morphological diagnosis, in particular, a fine needle aspiration biopsy.5. If the nature of the lesion is questionable, after 3-4 months of dynamic observation, it is necessary to repeat and expand the diagnostic search , 18.Thus, ultrasound examination and computed tomography play a leading role in the topical diagnosis of adrenal tumors. In the case of a large-sized tumor, angiography is the most informative test which allows determining the correct topical diagnosis in 92.6 % of cases. As an additional refining method in challenging cases, magnetic resonance imaging can be used.Positron emission tomography is a relatively new method of instrumental diagnostics, which is based on the fact that some radiopharmaceuticals at elevated adrenal function are more likely to exhibit enhanced cellular activity than standard imaging techniques. These drugs can be directed to individual enzymes that are formed in the cells of the adrenal tumors. For example, the use of 18F-dopamine allows capturing high-resolution images and detect malignant tumors.According to N. Ghanem et al., the use of this method allows differentiating benign from malignant lesions with high sensitivity and specificity – up to 100% . The litScintigraphy with an iodine-containing radiopharmaceutical makes it possible to determine the localization of various tumors of the adrenal glands with a diameter of 0.5 cm, as well as to detect diffuse or diffuse nodular hyperplasia of both glands in different types and subtypes of hyperaldosteronism. The essence of the method is that for the study, one can use a radioactively noticed predecessor of hormones of the adrenal cortex hormones – cholesterol. For this purpose, 131J-methylnorcholesterol (NP-59) was selected , 20.On the basis of the analysis of data on the inclusion of cholesterol in the synthesis of hormones, one can estimate the localization and functional characteristics of tumors in the cortical layer. Asymmetric accumulation of the radionuclide in the tissue of both glands allows identification of aldosterone-producing adenoma. In bilateral hyperplasia of the adrenal glands, the moderate absorption of radiopharmaceuticals by both glands is indicated in 72–120 hours.According to Di Martino et al., the sensitivity and positive predictive value of radioisotope scintigraphy were 90.9 and 83.3%, respectively. The evaluation of the efficacy, based on postoperative blood pressure monitoring, showed that the sensitivity and positive predictive value of the method were 91.6%, respectively. However, its specificity, including the diagnosis of unilateral small- and large-focal nodular hyperplasia, is approaching 100 % .As a variant of scintigraphy, before its application, inhibition of the secretory function of the adrenal cortex by dexamethasone (dexamethasone suppression test) is used. In this case, the image of the adenoma remains visible due to its increased secretory activity, and the image of hyperplastic tissue disappears. It is believed that in some cases, the use of the NP-59 test for the diagnosis of aldosterone-producing adenoma may replace the adrenal venous sampling test .Establishing the nosological form of hyperaldosteronism and distinguishing its subtypes is a critical step in the diagnosis.Traditionally, primary hyperaldosteronism is classified into four main subtypes, three of which are based on pathology , and the fourth is genetically predetermined. There are four genetic subtypes of familial hyperaldosteronism. With the deepening of knowledge about hyperaldosteronism, it became apparent that such a division is a gross simplification, as confirmed by morphological studies, which indicate a large variety of morphological characteristics of primary hyperaldosteronism.The aldosterone-producing adenoma is usually unilateral, but it may be bilateral, and its cellular composition varies from the cells of zona fasiculata to the cells of zona glomerulosa, the so-called ‘hybrid cells’. In turn, as a rule, hyperplasia is bilateral, but it may be either unilateral, diffuse, micronodular or macronodular. Also, it was established that aldosterone-producing adenoma, except solitary lesions, may be associated with diffuse or nodular hyperplasia within the residual ipsilateral cortex . From thConsequently, given these data, from a practical and clinical point of view, the differentiation of subtypes has the purpose of not trying to determine the morphological type, but the separation of bilateral, benign lesions with excessive unilateral production of aldosterone from malignant ones. Thus, it is necessary to find out which of the four subtypes of primary hyperaldosteronism is present in a single patient. The tasks of this stage are performed with the help of morphological diagnosis, genetic tests, adrenal venous sampling with measurement of aldosterone and cortisol levels.Determination of subtypes or differential diagnosis of subtypes of primary hyperaldosteronism begins with the exclusion of its rare form caused by aldosterone-producing carcinoma. Among the methods of morphological diagnosis, the cytological examination of punctate calcifications from the adrenal glands is most commonly used. Biopsy by needle puncture is ultrasound- or computed tomography-controlled. The results are used for differential diagnostics between nonspecific lesions of the adrenal glands  and tumors of the adrenal tissues . CytologGenetic testing is a component of differential diagnosis between the primary hyperaldosteronism subtypes. Familial forms of primary hyperaldosteronism are investigated using the polymerase chain reaction or other methods. It is recommended to investigate the mutation in the KCNJ5 gene in order to exclude or confirm the type III subtype  in childA dexamethasone suppression test is used when suspecting familial hyperaldosteronism type 1. In this case, despite the small doses (1-2 mg per day) of dexamethasone, there is a temporary decrease in the level of aldosterone in the plasma and urine, lowering the blood pressure .Adrenal venous sampling with measurement of aldosterone and cortisol levels for distinguishing unilateral from bilateral primary hyperaldosteronism is a mandatory recommendation according to the current diagnostic guidelines . This meHowever, in the case of patients under the age of 35 with a detected unilateral adenoma of more than 10 mm and a structurally unaltered opposite adrenal gland, adrenal venous sampling is not mandatory if the aldosterone-to-renin ratio is higher than 30 ng/dl (831 pmol/l) and hypokalemia is evident [adrenal venous sampling. In some centers, it is performed in all patients who had been diagnosed with primary hyperaldosteronism , 32. SomWith regard to the differential diagnosis of primary hyperaldosteronism subtypes, it is recommended to draw blood selectively from the central and segmental adrenal veins, with the subsequent determination of the concentration gradient of aldosterone and renin plasma at different levels of the venous bed of the individual adrenal gland .An essential indicator of selective blood collection is the lateralization index, which is the ratio of the concentration of aldosterone on the dominant side  to the concentration of aldosterone on the non-dominant side , 38. A lThe inclusion of adrenal venous sampling data in the differential diagnosis of primary hyperaldosteronism forms effectively reduces the risk of unreasonable adrenalectomy, which is performed only on the basis of computed tomography.The sensitivity and specificity of adrenal venous sampling for establishing the lateralization index are 95 and 100%, respectively, and 78 and 75% for computed tomography and MRI, respectively , 41. It It should be emphasized that the successes achieved in recent years in the topical diagnostics of adrenal tumors are due to the widespread use of ultrasound, computed and magnetic resonance imaging. At the same time, it should be mentioned that topical diagnostics and determination of the variant of primary hyperaldosteronism are sophisticated. Last but not least, their complexity lies in many morphological and genetic subtypes of the pathology, peculiarities of the morphological structure of aldosterone-producing adenoma, in particular, the abnormal presence of cells of zona fasiculata and glomerulosa. However, the accuracy of the diagnosis has a leading role in choosing the method of treatment  and in its long-term resultsThe authors confirm that there are no conflicts of interest.


Pleomorphic adenoma (PA) is the most common benign salivary gland tumor. When neglected on some occasions, its size and weight can enormously augment.We report a case of a giant submandibular pleomorphic adenoma in a 75 years old female patient which measured 34 × 26 × 20 cm and weighed 8.1 kg.Neglecting such tumors without treatment, can cause severe facial disfigurement and can even hinder the airway. According to Pinkston 1999, approximately 75%–85% of all PAs occur in the parotid gland, with only 8% arising in the submandibular gland. Those occupying the minor salivary glands represent 7–15% of all reported cases [PAs occur among all age groups, with the incidence rate of about 3.5/100,000. As the age progresses, the incidence rate escalates, but among older individuals; 65–74 years, a decline is noticeable. It is found more common among females . While aWe report this unique case to stress that such tumors can attain gigantic sizes and, if neglected and left untreated, could cause extreme facial disfigurement and also could affect the airways and compromise normal breathing. We report a case, which could be one of the largest recorded PAs of the submandibular gland.2A 75-year-old woman was referred to our hospital complaining of a huge swelling in the left side of her face and neck. She stated that this mass had begun to develop 15 years ago at which it was only the size of a small almond at the left side of her neck. The mass was painless and gradually increasing in size but the patient refused to get medical consultation throughout these years till she started to feel serious difficulty in breathing.Clinical examinations revealed a huge firm mass in the left side of the face and neck crossing the midline reaching the right side of the neck and inferiorly till reaching below the left inframammary sulcus. The mass itself was not tender, with nodular bosselated outer surface and the surrounding skin was congested in some areas , Fig. 2.The origin of the mass couldn’t be determined clinically whether it arised from parotid, submandibular or even thyroid gland. A core-needle biopsy suggested a salivary glandular origin.CT scan with contrast of the neck was done and revealed a huge mass (34 × 20 × 26 cm) arising from left side of the neck and extended to parapharyngeal and sublingual spaces with displacement of trachea to the right side. It was found separated from thyroid gland and surrounding structures. CT angiography was also done and showed that the main arterial supply was from the facial branch of the left external carotid artery, common, external and internal carotid arteries on both sided which were patent and of normal shape , Fig. 4.The patient was prepared for surgery. Anesthesia team used fibreoptic intubation. We started with a left inferolateral incision to separate the tumor from the neck great vessels. After securing them, we continued the dissection both superiorly and inferiorly in the medial direction to the right side , Fig. 6.The histopathology report grossly stated that the mass had lobulated irregular outer surface, and was covered by a skin flap measuring 30 × 10 cm which was grossly unremarkable. Cut section was heterogeneous showing yellowish friable areas admixed with whitish glistening areas, with wide areas of haemorrhage and cystic degeneration. Ten paraffin blocks were prepared. Microscopic analysis yielded a biphasic benign mixed tumoral proliferation formed of epithelial and myoepithelial components that were embedded against a myxochondroid matrix. The epithelial component comprised ductal structures lined by bland looking cuboidal cells. These were associated with clear plumpy, rounded epitheloid and plasmacytoid, as well as spindle shaped myoepithelial cells admixed with cartilaginous foci against myxoid background , Fig. 11The patient was discharged three days postoperatively in a good general condition with marvelous improvement of her respiration and facial appearance.To date, the largest ever recorded PA arising from the submandibular gland was 22 × 25 × 19 cm [3The incidence of giant PA is extremely uncommon, and it mostly arises from the parotid gland. It mainly occurs in females, with a mean weight of approximately 7.8 kg . Gupta eIt is extremely rare to report enormous Pas in the submandibular gland, only a few cases have been reported ,8. BeingThese tumors are usually painless, smooth, firm and asymptomatic until they attain major sizes mostly in the form of pressure symptoms. Imaging studies such as CT and/or MRI can give a reasonable diagnosis. It is usually well demarcated from the surrounding tissue by a pseudocapsule due to compression of the surrounding parenchyma and fibrosis. If this fibrous capsule is completely excised, these tumors could be cured with surgery. In huge sizes they could develop a variegated appearance with areas of hemorrhage, necrosis and calcification. Giant tumors commonly have a lobulated delineation, which supports the diagnosis.At the histological level, PAs demonstrate several phenotypes. Epithelial cells are arranged in sheets and islands showing typical ductal structures lined by epithelial cells with surrounding myoepithelial cells with heterogeneous features as spindle, squamous, clear, basaloid, oncocytic and sebaceous. The stroma characteristically is diverse with fibrous, chondroid, myxoid or hyaline aspects .The incidence of malignant transformation in PA ranges from 1.9% to 23.3% . The risRecurrence of such tumors is possible especially if the surgical margins are involved, that’s why it should be managed through complete excision of the tumor reaching completely free surgical margin.Presenting such cases highlights the problem of many patients especially in the developing countries who neglect such grave conditions which whatever the reasons should not be pardoned.4Giant PA of the submandibular gland is a rare finding, and should be diagnosed cautiously. The proper treatment is complete surgical excision reaching free surgical margins.There is no funding from any organization.Case reports are exempted from ethical approval in my 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.Mohamed Abdelkhalek: study design and writing.Mohamed Elmetwally: data collection.Alaa Mazy: data analysis.Mona Gad: data analysis.Shadi Awny: writing.Basel Refky: writing.Ahmed Abdallah: data collection.Amr F Elalfy: data analysis.Samar Abdallah: data collection.Abeer Elfeky: data collection.Mohamed A Hegazy: supervision.researchregistry5050.Mohamed Abdelkhalek.Not commissioned, externally peer-reviewed.There is no conflict of interests.