The specific pathogenesis of the diffuse large B-cell lymphoma(DLBCL)is still indefinite and argumentative. It is known that DLBCL is the most common type of non-Hodgkin's lymphomas (NHL). A lot of cases of DLBCL such as primary gastric diffuse large B-cell lymphoma(PG-DLBCL) are reported. However, primary intestinal diffuse large B-cell lymphoma(PI-DLBCL) is unusual.We present a case of a 57-year-old male diagnosed in the Gastroenterology Department, which presented a bleeding duodenal ulcer with irregular borders.The immunohistochemical staining showed: CD20(+++), CD10(+) and Ki-67>40%.The patient was successfully treated by Poly-chemotherapy with R-CHOP .After 6 courses of chemotherapy treatment, the duodenal ulcer was completely healed by reviewing the UGIE.Our report might give further strength to avoiding the erroneous and missed diagnosis for PI-DLBCL which is different from common duodenal ulcer. Although the primary location of diffuse large B-cell lymphoma is different, it has been reported that the proportion of diffuse large B-cell lymphoma in gastrointestinal lymphoma is 50% to 60% in China. But primary intestinal diffuse large B-cell lymphoma is rare. Moreover, the incidence of primary intestinal diffuse large B-cell lymphoma (PI-DLBCL) is rarely reported. However, studies about PG-DLBCL, primary mediastinal DLBCL, primary central nervous system DLBCL, paraneoplastic erythroderma, and primary nasal DLBCL all are reported.–6 The mechanism of action of DLBCL remains indefinite. The complications of DLBCL are also diverse and there is no unified standard. In this report, the patient was successfully treated by poly-chemotherapy with R-CHOP .Diffuse large B-cell lymphoma is the most common type of Non-Hodgkin lymphomas (NHL) in adults which accounts for 30% to 40% of NHL.2A 57-year-old man who presented with 20 days history of black color stool without treatment was taken to the hospital. The patient had upper abdominal pain, abdominal distension, and dizziness. But there were no other digestive syndromes containing acid reflux, heartburn, and vomiting. The patient had no previous medical history. Furthermore, there were no positive results by routine laboratory tests but exception of hemoglobin, 87 g/L and percentage of lymphocyte, 13.60%. In order to distinguish the nature of black stool, occult blood test is used because patients sometimes advocate taking bismuth drugs. And the stool occult blood test of this patient was positive. We took him for an upper gastrointestinal endoscopy (UGIE) examination that showed the following: a 1.5 × 1.5-cm and big ulcer of descending part of duodenum with irregular border. Congestion and edema existed around the mucosa of the duodenal ulcer. The surrounding mucosa is hypertrophied and devitalized, which differentiate it from peptic ulcer disease . Therefore, we took him for total abdomen Computed Tomography that showed a surprising result which was that the patient had a huge mass in the right mid-upper abdomen, which was considered to originate from the duodenum. So we considered this ulcer as a duodenal neoplasm. Afterwards, we were going to invite surgeons to consult patients to consider whether the patient should be treated with surgical treatment. However, the result of endoscopic biopsy came to our hands, which contained diffuse proliferation and infiltration of small blue cells which were found in the descending duodenum. Furthermore, immunohistochemical staining showed: CD20(+++), CD10(+), and Ki-67 >40%. However, we did tests including carcinoembryonic antigen, alpha fetoprotein, Carbohydrate antigen199, Cancer Antigen 724, ferritin, Cancer Antigen 242, Cancer Antigen 125, and prostate specific antigen, and they were all negative. And all observations showed that PI-DLBCL was identified . NHL lesions mainly occur in lymph nodes, spleens, thymus, and other lymphoid organs. They can also occur in lymphoid hematopoietic malignancies of lymphoid tissues. report that combination of Rituximab and Lenalidomide is an effective therapy for the patients with PG-DLBCL. And our report also shows the effectiveness of poly-chemotherapy with R-CHOP which contributes to improvement in curing PI-DLBCL is very obvious.Nowadays, although it is sometimes difficult to distinguish between benign and malignant ulcers, our report is based on reliable pathological findings with definite diagnosis. Moreover, if we can perform chromoendoscopy or NBI-magnifying chromoendoscopy, we may be able to detect the malignant tumor early. Although we initially suspected duodenal cancer, we were able to correct the diagnosis in time. Therefore, it requires our clinicians to perform a large number of tissue-depth biopsies of ulcers which could be suspected of being tumors. According to the literature report, Cai et alAccording to our report, we could put forward diagnostic criteria and therapeutic principles available for PI-DLBCL as follows: syndromes should include abdominal pain, abdominal distension, abdominal discomfort, or gastrointestinal bleeding; CD20(+) and Ki-67(+) should be detected from immunohistochemical staining; the treatment of proton pump inhibitor is ineffective; adenocarcinoma of duodenum should be excluded; R-CHOP chemotherapy is recommended as the first choice for treatment.In conclusion, we need more case reports and a long-term follow-up to make a greater contribution for diagnosis and treatment of PI-DLBCL.Conceptualization: Ning Jia.Visualization: Yanping Tang, Yang Li.Writing – original draft: Ning Jia.Writing – review & editing: Ning Jia, Yang Li.Ning Jia orcid: 0000-0002-5394-9004.


Imperforate hymen, though a congenital anomaly, usually presents late in puberty as lower abdominal pain, primary amenorrhea, and cyclical pain. Blood collects in vagina and uterus, proximal to imperforate hymen leading to their distention. Its presentation at infancy is a rare entity. We report such a rare case of symptomatic imperforate hymen in infancy, who presented with acute retention of urine, chills and rigor. Abdominal examination revealed an intra-abdominal mass in the lower abdomen and pelvis with the absence of vaginal opening on perineal examination. Contrast-enhanced computed tomography abdomen showed large abdominopelvic cystic lesion posterior to the urinary bladder and anterior to the rectum consistent with a highly distended vagina. She was managed by the incision of the imperforate hymen and drainage of the pus. A high index of suspicion is necessary whenever a female infant presents with abdomino-pelvic mass with symptoms of fever or urinary retention. The worldwide incidence is estimated at 0.014-0.1%. It is usually asymptomatic until menarche with an average presentation age of 11-15 years. It is rarely diagnosed in the neonatal period.3failure.A 4-month-old female infant presented with acute retention of urine and chills and rigor for 2 days. On examination, she was of normal weight and height for age. General physical examination was normal except for pyrexia. An abdominal examination revealed an intra-abdominal mass-like lesion in the lower abdomen and pelvis. There was no vaginal opening on perineal examination. The urethral and anal opening was normal. No other abnormalities were detected on systemic review.Contrast-enhanced computed tomography (CECT) abdomen showed large well-defined abdominopelvic cystic lesion posterior to the urinary bladder and anterior to the rectum in the rectovesical pouch; measuring about 14.0cm × 6.7cm × 6.3cm  × transverse (TR) × anteroposterior (AP)) consistent with a highly distended vagina. Superiorly it was communicating with a cystic tubular structure, which represented displaced and mildly distended uterus.Bilateral hydronephrosis and proximal hydroureter are also observed . The thiThe cruciate incision was made on hymen. Approximately 500ml of foul-smelling pus was drained. The postoperative period was uneventful. The patient was discharged on postoperative day 2. On 6 weeks follow up, the vagina and uterus regressed in size by ultrasonography . The operd of the vagina and the urogenital sinus which forms the lower 1/3rd. Usually, hymen ruptures before birth due to degeneration of central epithelial cells.4 Imperforate hymen occurs due to abnormalities in the degeneration of the central epithelial cells. This may result in hydrocolpos which is cystic dilation of the vagina with fluid accumulation proximal to obstruction due to stimulation of secretory glands of the reproductive tract and maternal estrogen.5 On examination of the genitalia of a newborn, it can be appreciated as a bulging membrane. Following this, there is the reabsorption of the mucus and the child becomes asymptomatic only to present with various symptoms after menarches such as primary amenorrhea, cyclical abdominal pain or urinary retention.6The hymen is a membranous structure lying between the vagina and the vulva. Embryologically, it corresponds to the junction of the caudal end of the Mullerian duct which forms the upper 2/37,8The complications of congenital hydrocolpos include urinary tract obstruction due to compression, renal failure, repeated urinary tract infections, rupture, and peritonitis. Sepsis leading to death can occur secondary to either urinary tract infection or rupture and secondary peritonitis.7These complications can be managed by simple hymen incision and drainage of the collection. Therefore, a thorough newborn examination is essential to screen for an imperforate hymen. Magnetic resonance imaging is especially helpful when hydrometrocolpos is secondary to cloacal malformation. Magnetic resonance imaging is also preferred when ultrasonography is limited by the patient’s obesity or oligohydramnios in suspicious antenatal scans.Neonatal hydrocolposis a rare condition and occurs secondary to vaginal obstruction and stimulated cervicovaginal secretions. A high index of suspicion is necessary whenever a female infant patient presents with abdomino-pelvic mass with symptoms of fever or urinary retention. Diagnosis can be confirmed using ultrasonography, computed tomography, or magnetic resonance imaging, with MRI being of particular use in the presence of other cloacal abnormalities due to its superior soft-tissue resolution. Early diagnosis reduces the incidence of complications such as infection, rupture, and renal failure.Consent: JNMACase Report Consent Form was signed by the patient and the original is attached with the patient chart.None.


Lipomas are common benign tumors, constituting 16% of soft tissue mesenchymal tumors. They usually occur under the skin or in the large muscles of the thigh, shoulder, or upper arm. There are few reported cases of lipomas located in the forearm and hand muscles, accounting for less than 1% of all lipomas.A 47-year old man presented with a history of swelling and accompanying pain in the left wrist for 2 years.The patient was diagnosed with intramuscular lipoma in the pronator quadratus.The mass was resected completely with wide-awake technique.The patient was followed up for 2 years with no recurrence. The symptoms of swelling and pain resolved within 3 weeks post-surgery, and there was no clear abnormality in wrist and finger movement and sensation. A satisfactory outcome was achieved.Intramuscular lipoma in the pronator quadratus is a rare benign tumor which should be distinguished from malignant tumors. Especially for patients with carpal tunnel syndrome presenting with wrist swelling, ultrasound, computed tomography, or magnetic resonance imaging can be used to assess deep tissue masses. There are rarely reported cases of lipomas located in the forearm and hand muscles,,2 accounting for less than 1% of all lipomas. In this paper, we present the case of a lipoma in the pronator quadratus. No recurrence was observed in this case in 2 years of follow-up.Lipomas are common benign tumors, constituting 16% of all soft tissue mesenchymal tumors.2A 47-year-old man presented with a 2-year history of swelling and accompanying pain in the left wrist, but no paresthesia. Massage and infrared treatment had resulted in no obvious improvement in these symptoms. Ultrasound examination revealed a 4.0 cm × 3.0 cm subcutaneous hypoechoic mass with no blood flow. Physical examination indicated no swelling of the superficial lymph nodes. There was an intumescence at the distal end of the left forearm, with no dark purple skin Fig. A. On pal was used for mass resection: a 6-cm arc-shaped incision was made at the ulnar edge of the long palmar muscle of the distal forearm, followed by peeling back the skin and subcutaneous tissues  account for less than 1% of all lipomas. In 1853, Paget reported the first case of IML, followed by several reports of IMLs mostly located in the thigh, shoulders, and upper arm.–10 IMLs of the forearm and hand are relatively rare, and have been reported in the thenar muscle and the thumb.,11,12A lipoma is the most common benign soft tissue tumor arising from mesenchymal tissues and is composed of mature adipose cells. Lipomas can occur at any age, but are most often seen in the 40 to 70 year old population, without gender preference. found that two-thirds of patients with lipomas are overweight or obese, so the occurrence of IML may be related to increased body mass index. Mori believed that the occurrence of IML may be related to local neurogenic or myogenic diseases. He found that selective muscle fiber atrophy or degenerative changes often occur in IMLs, most of which are invasive. This infiltration is thought to relate to abnormally elevated levels of mobility group proteins. Fibrous atrophy is not limited to the area of fatty infiltration but can also be found in peripheral muscle fibers involved with the tumors.As with other types of lipomas, the pathogenesis and infiltration mechanism of IMLs are unclear, and they may be solid tumors that originate directly from pluripotent mesenchymal cells. They are also thought to be associated with uncontrolled adipose growth caused by trauma, chronic irritation, obesity, developmental disorders, endocrine and metabolic disorders, or genetic factors. In addition, Ramos-Pascua et al When an IML is located in the posterior pharyngeal space, the patient may develop difficulties in swallowing and speech due to recurrent laryngeal nerve and supra-larynx nerve compression. When an IML is located under the tongue, it may cause irritation and difficulties in swallowing and speech.,17 An IML in the chest may even be misdiagnosed as breast cancer. Therefore, intramuscular lipomas are often misdiagnosed as malignant tumors. An IML in the pronator quadratus, as in this case, could also cause a disorder of forearm rotation by compression of the terminal branch of the anterior interosseous nerve, or produce carpal tunnel syndrome due to traction of the median nerve.IMLs often manifest as an asymptomatic, slow-growing mass or swelling with no obvious mass. The clinical manifestations are mainly determined by the position of the tumor growth. When the tumor is large enough to invade the muscle, limitation of motion may appear. When the peripheral nerve is pressed, it may cause paresthesia but generally does not cause pain. In this case, the initial diagnosis was only based on the ultrasound results, and no further examination was performed to re-confirm the nature and location of the tumor. In general, we would caution implementing a surgical exploration, by which the tumor is prone to be misdiagnosed or missed, without thorough diagnostic imaging.Imaging examination is the most important tool for the diagnosis of IML. Ultrasound is the first choice for its convenience, speed, and low cost. It can dynamically show the relationship between the location and depth of the tumor and the adjacent tissue. Under ultrasound, IMLs appear as intramuscular echogenic masses with clear borders and no blood flow signals, in which the echo intensity is higher than that of the muscle but similar to subcutaneous adipose tissues. The tumors encircled by muscle fibers can be heterogeneous and have internal visible streaks. Computed tomography (CT) and magnetic resonance imaging (MRI) can more clearly reveal the integrity of the capsule, the fat content in the tumor, the degree of fat and muscle, and the thickness and distribution of fiber intervals. Intramuscular low-density masses can be seen on CT; both high T1 and T2-weighted signals are observed on MRI. Fat suppression MRI sequencing is an available approach to comprehensively and clearly assess peritumoral edema and the degree and range of tumor invasion to the surrounding muscle tissues. It is also useful in identifying other tumors, and especially malignant tumors. Histopathological examination is the main approach to distinguish IML from well-differentiated liposarcoma. IMLs manifest with mature and single-celled adipocytes of equal size and uniformity, with occasional muscle fiber bulbs. The adipocyte nuclei are small, flat, and located peripherally, without cellular atypia, mitotic activity, polykaryon, or lipoblasts. In contrast, liposarcoma is a mixture of atypical cells or vacuolarized adipocytes with spindle-shaped fibroblast-like cells, often located in many thick intervals, with vascular components of varying sizes.It is not difficult to diagnose IML based on symptoms, signs, and imaging examinations. However, most IMLs are invasive, and need to be differentiated from muscle spasm, hemangioma, liposarcoma, fibromyositis, and fibrosarcoma. IMLs can be divided into infiltrative, well-defined and mixed type. For well-defined masses, marginal resection is preferred. In infiltrating masses, muscle fiber bundles pass through the tumor, and a small number of IMLs are enlarged through other structures or surroundings, which necessitates expanded surgical resection. If complete resection is not possible or complete resection can lead to severe dysfunction, partial resection may be adequate. When the origin of the mass cannot be determined, but there is a secondary infiltration of adjacent muscles, a small amount of surrounding tissue can be removed. When it is difficult to make a judgment, preoperative puncture biopsy for pathology examination can be used to distinguish from other malignant tumors such as low-grade liposarcoma, potentially avoiding unnecessary expanded resection. The excised intramuscular lipoma is composed of localized, uniform yellow adipose tissues, which is a somewhat brownish color, usually without a capsule but with a soft texture. There are usually no signs of necrosis or bleeding visible to the naked eye.Generally, asymptomatic IMLs can be assigned observation status, and the recurrence rate after surgical removal is less than 5%.4,23In summary, IML in the pronator quadratus is a rare benign tumor which should be distinguished from malignant tumors. Especially for patients with carpal tunnel syndrome presenting with wrist swelling, ultrasound, CT or MRI can be used to differentiate IMLs from other deep tissue masses.All authors substantially contributed to the manuscript. TH and QWR designed the study. PJB and LJ performed the literature review, extraction of the data, and analysis of the pooled data. TH and ZZ performed surgical resection and perioperative care. LR reviewed and revised the manuscript. All authors read and approved the final manuscript.Data curation: Wenrui Qu, Zhe Zhu.Formal analysis: Wenrui Qu.Investigation: Heng Tian, Wenrui Qu, Zhe Zhu.Project administration: Rui Li.Resources: Zhe Zhu, Jun Liu.Supervision: Jun Liu.Writing – original draft: Heng Tian, Junbo Pan.Writing – review & editing: Rui Li.


Primary lymphomas of the salivary glands are rare tumors seen in the head and neck. We report the case of a 52-year-old woman with an extranodal marginal cell lymphoma of her submandibular gland, whose primary presenting symptom was a slow swelling of her neck. Fine-needle aspiration of the mass was performed, followed by CT imaging studies, and both tests showed inconclusive results. The tumor was excised and the patient has been monitored with no recurrence to date. This case adds to the diverse differential diagnoses of primary tumors in the head and neck, as early consideration, identification, and management of this etiology is essential for the clinician to ensure the best patient outcomes. Neoplasms of the salivary gland are rare entities, representing 10% of head and neck tumors . ExtranoA 52-year-old woman presented with an asymptomatic, slowly growing swelling of the left neck. She had undergone an ultrasound, which showed a 1.5 x 0.7 x 1.1 cm hyperechoic mass inside of the left submandibular gland, prompting her to visit an otolaryngologist. Fine-needle aspiration of the mass was performed in the office, which revealed lymphocytes and benign epithelium. Given these results, the differentials for the mass included sialadenitis and lymphoid lesions. She subsequently underwent a CT soft tissue of the neck, which failed to demonstrate the submandibular gland abnormality seen on ultrasound. A decision was made to proceed with surgery to excise the submandibular mass. Final pathology post-excision showed normal salivary gland tissue contrasted with abnormal salivary gland tissue with significant lymphoid infiltrate Figure . These oExtranodal marginal cell lymphoma can affect any extranodal mucosal tissue, with the most common tumor sites being the stomach, other gastrointestinal organs, skin, and respiratory tract . The salThe treatment protocol for submandibular gland marginal cell lymphoma remains a matter of minimizing long-term treatment-related complications and hence should be individualized to the patient’s specific clinical picture. This represents a diagnostic distinction from most submandibular primary malignancies, in which surgical resection of the tumor with a potential need for neck dissection and/or adjunctive radiation therapy is the first-line treatment. It is important for otolaryngologists to be mindful of this pathological entity in patient counseling and determining treatment regimens regarding submandibular neoplasms.


Acquired Corneal Sub-Epithelial Hypertrophy (ACSH) has been described in patients with peripheral superficial corneal opacities following penetrating keratoplasty and might present similar to Salzmann’s nodular degeneration (SND) or peripheral hypertrophic sub-epithelial corneal degeneration (PHSCD). We describe the clinical presentation, topographic findings and the surgical outcome of three cases, which fit the appearance and characteristics of ACSH.Three patients (3 eyes) with paracentral or peripheral corneal opacification were reviewed to describe their clinical examination (SL), morphology of the opacity  and document their topographic changes before and after surgical intervention by peeling of the epithelium with or without superficial keratectomy under the microscope in addition to brief description of their histopathological examination.All 3 cases were secondary to corneal procedures [Penetrating keratoplasty (PKP) in 1 for pseudophakic bullous keratopathy and deep anterior lamellar keratoplasty (DLK) in 2 for advanced keratoconus]. All cases presented with reduced vision, astigmatic changes in topography or manifest refraction. The visual acuity, symptoms, and topographical findings all improved after treatment. Histopathologically, all cases fit the newly described entity of ACSH.Careful clinical judgement guided by corneal topography are needed for proper the diagnosis of acquired corneal opacification that results in reduction of vision to identify ACSH from other similar conditions (PHSCD and SND). Peeling of the thickened epithelial and sub-epithelial tissue is curative avoiding the need for corneal re-grafting. It is typically bilateral, symmetrical, peripheral, and sub-epithelial in location ,2. This 2This case series was prepared in accordance with the ethical standards of the human ethics in accordance with the Helsinki Declaration. No IRB approval is needed for case reports. However, a general informed consent was taken from all patients, which includes permission for anonymous reporting. The authors have no financial disclosures to declare nor conflict of interest in relation to this case series. No financial funds involved. Research registry of the case series: #researchregistry5315. We retrospectively report three cases with identical clinical and histopathological features to their described entity, who were similarly treated with superficial corneal peeling in 2 centers: King Abdulaziz University hospital at King Saud University (two cases) and King Khaled Eye Specialist Hospital (one case). Both centers are government academic tertiary eye care hospitals. The surgical procedure was performed under topical anaesthesia in each corresponding center where the patient originally presented by the primary caring consultant ophthalmologist, all of whom had long experience in the field of cornea and external disease and are included as co-authors. The obtained specimens were histologically examined by the senior author for confirmation of the specific entity of ACSH. The follow-up periods for the patients have been variable according to their initial presentation date however, they all share a common satisfactory post-operative result. No long-term follow-up was necessary in these cases since a reasonable clarity of the cornea was shortly obtained after the surgical intervention.33 consecutive cases have been included with the following description of each case.3.1A 36-year-old gentleman who is known to have advanced keratoconus for which deep anterior lamellar keratoplasty was done in his left eye for visual rehabilitation. After removal of all sutures and satisfactory period of several years, during which his uncorrected vision (UDVA) was measuring 20/30 in his left eye, the patient started to notice a progressive blurring of vision. At presentation, his left eye UDVA was 20/80 improving with pinhole to 20/40. The intraocular pressure (IOP) was 13 mmHg. The conjunctiva was quite but the cornea was showing an arcuate opacity around the suture-less edge of the graft temporally resembling a corneal keloid like picture with a clear center a. The riThe surgical intervention was performed by simple gentle manual peeling of the opacified thickened epithelium and sub-epithelial tissue aiming at achieving a clear and smooth stromal plane without causing any surface irregularity followed by application of Mitomycin C (MMC) 0.02% for 2 min with copious irrigation of the corneal surface with Balanced Saline Solution (BSS). After the procedure bandage contact lens was applied to allow corneal epithelial defect to heal and to ease the post-operative recovery for the patient under the coverage of prophylactic broad-spectrum antibiotic drops as well as steroid drops to prevent the theoretical risk of recurrence from the robust wound healing process. The excised tissue showed variably thickened corneal epithelium with no Bowman’s layer and sub-epithelial fibrous tissue c. There One week following the procedure, the patient showed an improved measured visual acuity I the left eye: (UDVA: 20/40). The epithelial defect healed with significant disappearance of the corneal opacity and better graft clarity d. Repeat3.2A 78-year-old male was referred to the cornea service at King Khaled Eye Specialist Hospital four years prior to this recent presentation as a case of Pseudophakic Bullous Keratopathy (PBK) in the left eye with Best corrected visual acuity (BCVA) measuring 20/300. He had Penetrating keratoplasty (PKP) and his BCVA improved to 20/60. Four years later, his BCVA dropped to 20/200 and his left eye slit lamp examination a showed 3.3A 34-year-old- male patient, is a known case of advanced keratoconus in the left eye, for which he underwent deep anterior lamellar keratoplasty (LK) 2 years ago. The patient had quiet post-operative course and all sutures were removed 8 months after the surgery with BCVA of 2030 but eventually his vision started to decrease to a BCVA of 20400 at presentation. Upon examination, he was found to have quiet conjunctiva. Examination of the cornea showed suture-less corneal graft with sub-epithelial non-vascularized elevated corneal opacity that was more prominent in the temporal aspect of the graft measuring about 2.5 × 3 mm with faint smooth borders. The nasal aspect of the opacity was less dense compared to the temporal one with sparing of the visual axis. Otherwise, the graft was relatively clear with no evidence of underlying edema a.Topographically, there was an irregular corneal surface on the sagittal curvature map due to massive flattening effect caused by the corneal opacity temporally with total anterior astigmatism of 16 diopters, also there is evident anterior elevation corresponding to the area of opacity. on pachymetry map there was marked thickening at the area of the opacity temporally b.The patient was counseled regarding the surgical option for visual rehabilitation of his condition and he was offered simple peeling in the minor operating room under topical anesthesia, which was done in a similar fashion as in case 1 with no complications. The excised tissue was identical to the other 2 cases with thick irregular corneal epithelium, absent Bowman’s layer and sub-epithelial fibrosis but there was a small focal area of degenerative non-inflammatory pannus c and d. 4Acquired Corneal Sub-epithelial Hypertrophy (ACSH) is a distinct histopathological corneal entity which can affect the vision due to irregularity in the corneal surface causing high irregular astigmatism and characteristic topographic findings in the form of an increased thickness and front elevation with normal back elevation . AlthougAs in any very superficial corneal lesion in symptomatic patient and when medical therapy alone is likely to fail, membranectomy by simple peeling may be sufficient (because bowman’s layer is not deeply involved) with or without superficial keratectomy , in which finding this plane is more difficult due to deeper involvement) ,6,7. MitIn our cases, superficial peeling was sufficient to achieve corneal clarity and improve vision.The advantage of this treatment is that it is a minor surgical intervention with a faster recovery. Recurrence was not noted for 3 months in both of our patients, but long-term follow up is needed to detect any recurrence in these patients even though recurrence of opacity is not expected. The major limitation of this case series was the rarity of this condition, thus the limited number of patients included. Also, the lack of definite explanation and exact pathogenesis for the development of this condition. This warrants further multicentric collaborative study in the future to collect enough number of similar cases.5We believe that ophthalmologists should be aware of this newly described entity in order to be able to identify cases of ACSH and treat them accordingly with simple peeling or superficial keratectomy saving them from the more invasive penetrating keratoplasty procedure. Further ultrastructural and biochemical studies are needed to clarify the etiology, pathogenesis, and long-term outcome behind development of this specific type of corneal opacity especially following LK or PKP for possible preventive measures.The Case series has been supported by King Saud University Medical City, however there were no funds involved.This case report was prepared in accordance with the ethical standards of the human ethics in accordance with the Helsinki Declaration. Case reports do not require IRB approval; however, the General Informed Consent includes patients’ approval for use of relevant clinical and surgical information in an anonymous way.General informed consent has been taken from all patients for use of relevant clinical and surgical information in an anonymous way.First and second authors: Collection of data, literature review, and drafting the manuscript.Third, fourth and fifth authors: Clinical diagnosis and surgical management of the patients.Last senior author: Critical review of the manuscript, histopathological diagnosis, and corresponding author.Registration has been obtained: #esearchregistry5315.Dr. Hind Alkatan.Not commissioned, externally peer-reviewed.The authors declare that they have no competing or conflict of interest, and the General Informed Consent includes patients’ approval for use of relevant clinical and surgical information in an anonymous way.


A 9-year-old male, castrated Chihuahua was examined because of a 7-day history of intermittent vomiting. A mass in the small intestine was identified on abdominal radiography and ultrasonography. Laparotomy revealed a mass lesion originating in the ileum, and surgical resection was performed. The mass was histologically diagnosed as adenosquamous cell carcinoma. Chemotherapy with carboplatin was initiated, but the dog was suspected to have experienced recurrence 13 months after surgery and died 3 months later. To our knowledge, this is the first case report to describe the clinical course of adenosquamous cell carcinoma in the small intestine of a dog. Lymphoma is the most common type of intestinal tumor in dogs, followed by adenocarcinoma, leiomyosarcoma, and gastrointestinal stromal tumor . AdenosqIn Japan, there is no ethics committee for private-practice veterinary hospitals. Nevertheless, this research was conducted according to the ethical guidelines of the Japan Veterinary Medical Association. The samples were collected after obtaining written consent from the dog’s owner.A 9-year-old male, castrated Chihuahua was brought to our veterinary hospital with a 7-day history of intermittent vomiting. Physical examination revealed a body weight of 1.90 kg and a body condition score of 1 ; the dog was extremely thin. The body temperature was 38.7 °C, heart rate was 150 beats/min, and respiratory rate was 48 breaths/min. His general appearance was quiet, alert, and responsive. Abdominal palpation revealed the presence of fecal impaction in the caudal abdomen.A hematological analysis indicated leukocytosis , 6000–17,000/μL) with neutrophilia  including 354 band neutrophils). Biochemical analyses revealed abnormalities, including an increased concentration of C-reactive protein (CRP)  and decreased concentrations of albumin  and total calcium . The in-house heartworm antigen test based on immunochromatography showed a negative result. Urinalysis revealed a specific gravity of 1.048 and weak proteinuria, and the urine protein/creatinine ratio was normal . Thoracic and abdominal radiography revealed no thoracic abnormalities; however, the caudal abdomen was unclear due to fecal impaction. Abdominal ultrasonography revealed a mass (approximately 2.5 cm in diameter) with mixed echogenicity. The mass was suspected to have originated in the intestinal tract. Fine-needle biopsy was not performed due to the distribution of many blood vessels within the mass. No intraperitoneal lymphadenomegaly was observed. Based on these results, the intestinal mass was considered to be the underlying cause of the severe weight loss and intermittent vomiting. Famotidine   was administered until the scheduled surgery date.On day 3, an additional blood test performed before surgery revealed normal total bile acid concentration , prothrombin time , and activated partial thromboplastin time  and a mild increase in fibrinogen concentration . Laparotomy was performed under anesthesia for resection of the mass. The mass originated from the ileum (3.5 cm × 3.0 cm × 2.5 cm). No intraperitoneal lymphadenomegaly and no peritoneal effusion were observed. Omental adhesions were observed in parts of the mass. The mass was macroscopically normal in color and appeared to have a thickened intestinal wall A. The poPostsurgical care included hospitalization and administration of lactated Ringer’s solution  , meloxicam  , enrofloxacin  , ampicillin  , and famotidine . On day 11, the dog was discharged following an improvement in vomiting condition compared with that on day 1.Histopathological findings revealed that in the ileal mass, the mucosal epithelium exhibited neoplastic proliferation. Tumor cell atypia was moderately strong or moderate to moderately strong depending on the site, with a mitotic index of 75 (per 10 high-power fields). Tumor cells proliferated while forming lobular structures, showing a tendency of keratinization. Areas were also present in which tumor cells grew while forming irregular glandular cavities and lumens. The tumor comprised both malignant squamous and glandular components B. Tumor 2, IV, every 3 weeks)  was initiated based on histopathological findings of tumor vascular invasion. By day 123, the dog’s weight had recovered to 3.0 kg (body condition score of 3), and no abnormalities were observed in blood chemistry analysis or thoracic and abdominal radiography and ultrasonography. Five rounds of carboplatin chemotherapy were completed and discontinued on day 123.By day 32, the dog’s weight had recovered to 2.1 kg, with good general condition. Albumin (2.6 g/dL) and CRP (<0.9 mg/dL) concentrations had also returned to within the RI. Thoracic and abdominal radiography and ultrasonography showed no abnormalities. On the same day, carboplatin chemotherapy  was observed. Thoracic radiography revealed nodular lesions in the right cranial lobe (approximately 2.0 cm in diameter) and the middle lobe of the right lung (approximately 1.0 cm in diameter) . AbdominASCC is defined as a carcinoma containing adenocarcinomatous and squamous cell carcinomatous components . The hisIn humans, reports of ASCC in the small intestine are extremely rare . In fourConsidering that histopathological findings showed vascular invasion, chemotherapy was performed in this case. As there are no reports in the literature regarding chemotherapy for ASCC in dogs, carboplatin was administered with reference to chemotherapy for intestinal carcinoma . There iIn this study, the CRP concentration was elevated at the time of diagnosis of ASCC. CRP is an acute-phase protein produced in the liver in response to an increase in the release of pro-inflammatory cytokines such as interleukin-1, interleukin-6, and tumor necrosis factor during inflammation . The CRPTo the best of our knowledge, this case report is the first to describe the clinical course of ASCC in the small intestine of a dog.


Primary mucinous tumors of the testis and paratestis are very rare, with only 29 reported cases detected in a PubMed search. The histopathological characteristics of primary testicular mucinous tumors are similar to their ovarian counterparts, and the diagnosis and naming criteria refer to the criteria for female ovarian mucinous tumors. However, the clinical and imaging features of primary testicular mucinous tumors are poorly understood, and they are thus frequently undiagnosed or misdiagnosed. We present the case of a patient with a primary testicular mucinous tumor. A 52-year-old man presented with a 1-year history of painless enlargement of the left scrotum. Ultrasound examination revealed a cystic mass in the left testis, with viscous fluid areas and calcified spots, irregular solid bulges on the cyst wall, and a small blood supply. Serum alpha-fetoprotein, β-human chorionic gonadotropin, lactate dehydrogenase, renal function, inflammatory markers, and routine urine and blood examinations were all normal. The patient underwent radical resection of the left testis. Postoperative pathology showed a multilocular cystic mass, with the inner wall of the sac lined with mucous columnar epithelial cells, some with mild nuclear atypia, and no interstitial infiltration. The pathological diagnosis was testicular mucinous tumor. Postoperative abdominal and pelvic computed tomography, colonoscopy, and gastroscopy showed no suspicious lesions. The final diagnosis was primary testicular borderline mucinous tumor. The patient underwent postoperative follow-up examinations once a year for 4 years. Serum tumor markers, scrotal ultrasound, abdominal and pelvic computed tomography scans, and colonoscopy and gastroscopy revealed no evidence of metastases or other primary adenocarcinoma. This case highlights the clinical and imaging characteristics of primary testicular mucinous tumors, which might aid their differential diagnosis. Primary tumors of the testis and paratesticular tissue include various pathological types. Germ cell tumors are the most frequent, while ovarian epithelial tumors are rare, and mucinous tumors are extremely rare, with only 29 reported cases detected in a PubMed search –3. The hA previously healthy 52-year-old man presented with a 1-year history of painless left scrotum enlargement and mild discomfort, but no lower urinary tract symptoms. He was admitted 1 month after his symptoms worsened, with subjective fever, but still without lower urinary tract symptoms. Before admission to hospital, he had been treated at a local community hospital and had received oral empiric antibiotic levofloxacin for 2 weeks, with no significant improvement in his symptoms. The patient had no history of cryptorchidism, scrotal trauma, scrotal inflammation, or urinary tuberculosis, and no history of neoplastic disease or family history of genetic diseases.Physical examination of his testes revealed an enlarged, hard, and non-tender left testis, a clinically normal right testis, and no palpable inguinal lymphadenopathy. Ultrasound examination of the scrotum revealed a mainly cystic, heterogeneous echogenic mass occupying most of the left testicle. The tumor had a clear boundary, regular morphology, and disordered internal echo  and upper and lower gastrointestinal tract endoscopy, but no other suspicious primary tumors or metastases were found. The tumor was thus finally diagnosed as a primary borderline mucinous tumor of the testis, according to the 2016 edition of the WHO Classification of Tumors of the Urinary System and Male Reproductive Organs , while teratomas rarely occur in patients over 40 years old (median 23–29 years old) –32. Path tubules , 2. The Reports of treatment experiences for patients with primary testicular mucinous tumors are limited, and there are currently no clear treatment guidelines. Radical orchiectomy is the most commonly used treatment –25. AmonIn conclusion, the analysis of currently reported cases indicated that primary mucinous testicular tumors tend to occur in people over 40 years old, with nearly a third of patients over 40 years old having malignant tumors; however, their clinical manifestations and tumor markers lack specificity, and the predominant imaging characteristics include a predominantly cystic mass in the testis and adjacent tissues. Metastatic testicular mucinous tumors and teratomas are difficult to distinguish based on their imaging characteristics, and abdominal and pelvic CT examination and gastrointestinal endoscopy are needed to exclude metastatic mucinous tumors. The final diagnosis depends on histopathological examination. Radical orchiectomy is the most widespread treatment for primary mucinous tumors of the testis, and scrotal ultrasound, abdominal and pelvic CT, and gastrointestinal endoscopy should be performed and reviewed regularly after surgery.The original contributions presented in the study are included in the article/The studies involving human participants were reviewed and approved by appropriate institutional of Shanxi Bethune Hospital. The patients/participants provided their written informed consent to participate in this study. Written informed consent was obtained from the individual(s) for the publication of any potentially identifiable images or data included in this article.CH, ZY, and XK wrote the manuscript. CK revised the manuscript. TL and JX reviewed the manuscript. All authors contributed to the article and approved the submitted version.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.


Ganglioneuromas are slow-growing hamartomatous tumors that are rarely found in the colon. There are three subtypes of ganglioneuromas: polypoid ganglioneuroma, ganglioneuromatous polyposis, and diffuse ganglioneuromatosis. They are differentiated depending on their endoscopic and histological characteristics. Patients with colonic ganglioneuroma may present with nonspecific symptoms; however, they are usually asymptomatic. We present a case of hematochezia, where an ascending colon ganglioneuroma was found incidentally on diagnostic colonoscopy. We will explain how to distinguish the three subtypes of ganglioneuroma on colonoscopy and will mention the genetic disorders associated with them. We will also discuss the treatment of ganglioneuromas. Ganglioneuromas are slowing-growing tumors that are rarely found in the colon. There are three subtypes of ganglioneuromas: polypoid ganglioneuroma, ganglioneuromatous polyposis, and diffuse ganglioneuromatosis . They arAn 84-year-old male patient with a past medical history of hypertension, chronic atrial fibrillation on rivaroxaban, chronic obstructive pulmonary disease, and prostate cancer status post-radiation presented to the hospital with hematochezia of two-day duration. He had three loose and bloody bowel movements on the day of presentation, with bright red blood and clots present. Hematochezia was associated with nausea and diarrhea. However, the patient denied abdominal pain, vomiting, hematemesis, melena, or weight loss. Last colonoscopy around seven years ago was normal. Vital signs were within the normal limits. Physical examination showed an irregularly irregular rhythm. Abdominal examination was unremarkable. Rectal examination showed dark red stools without any external hemorrhoids. Routine labs were significant for a drop of hemoglobin from 12.5 g/dL to 11 g/dL. International normalized ratio was 1.4. Colonoscopy was performed five days after stopping Xarelto®. It showed a 3-mm polyp in the ascending colon (Figure Ganglioneuromas are hamartomatous tumors that usually occur in the head, neck, or adrenal glands. They are rarely seen in the gastrointestinal tract. They originate from undifferentiated neural crest cells, although the exact mechanism of their formation is unknown. It is hypothesized that a soluble nerve growth factor may contribute to their development . There aPatients with colonic ganglioneuromas can be either asymptomatic or present with abdominal pain, hematochezia, constipation, diarrhea, ileus, weight loss, or obstruction. The presence of these symptoms and their severity depend on the size, location, and subtypes of the lesions. On endoscopy, the different subtypes of ganglioneuromas are distinguished based on the number and size of the polyps. Polypoid ganglioneuroma can be either a single lesion or a few lesions less than 2 cm in size. It is not possible to differentiate them from hyperplastic or adenomatous polyps by endoscopy, and, as a result, diagnosis can only be made through histological examination. Ganglioneuromatous polyps are more numerous. They are usually greater than 20, but each polyp is less than 2 cm in size. Diffuse ganglioneuromatous are large, ranging from 1 to 17 cm in diameter. They usually spare the ileum and may distort surrounding tissue. Ganglioneuromas can be associated with genetic disorders depending on their subtype. Single lesions such as polypoid ganglioneuroma are usually an isolated finding. They are not associated with any genetic syndrome. Patients with ganglioneuromatous polyposis can have associated anatomical findings such as several skin tags or cutaneous lipomas that can be present either on the face or on the extremities ,4. They Treatment of colonic ganglioneuromas depends on their size, location, and subtype, as well as the patient’s clinical history. Treatment also depends on the presence of complications such as bleeding or obstruction. Polypoid ganglioneuroma is a benign lesion; therefore, endoscopic resection using a hot biopsy forceps is usually curative. Although there are not any recommendations on how to further manage polypoid ganglioneuroma, it may be useful to perform a follow-up colonoscopy to ensure complete excision of the lesion . The treDefinitive diagnosis of ganglioneuroma can only be made through pathological and histological examination. Histologically, ganglion cells are seen mixed with proliferation of Schwann cells. Hematoxylin and eosin staining will show multiple spindle cells, which is usually enough to confirm the diagnosis. Usually, these lesions are immunoreactive to neuron-specific enolase and S100 protein stain, which is helpful in confirming the neural origin of the legion . There aIn summary, colonic ganglioneuromas are very rare. We presented a case of a patient who presented to the hospital with hematochezia. An ascending colon polyp, which turned out to be a ganglioneuroma, was found incidentally on colonoscopy. Bleeding may have been caused by the ganglioneuroma or may be unrelated.


A 50-year-old woman presented with a 5-month history of recurrent urinary tract infections. She had no complaints of any intestinal symptoms. She had been treated previously with oral antibiotics. The episodes became more frequent and she started with pain in the lower abdomen and fetid urine. Complete study lead to diagnosis of adenocarcinoma of the appendix with bladder fistula. The lesion was removed by laparoscopic right hemicolectomy and en bloc partial cystectomy. Pathological examination revealed a mucinous adenocarcinoma that had originated in the appendix and extended into the bladder wall. Six years after the operation, the patient remains asymptomatic with no evidence of recurrent or metastatic disease. Appendiceal carcinoma extending to the bladder is extremely rare and approximately 40 cases have been described. Management of recurrent urinary tract infections should not limit to empiric antibiotic therapy before the exclusion of possible organic causes. Appendiceal carcinoma may invade the bladder without intestinal symptoms but with urinary symptoms only, because of its anatomical position. The recommended treatment for non-carcinoid appendiceal tumours is right hemicolectomy and for T4 tumours en bloc resection of the involved structures. Further study is needed to determine adjuvant therapy. A literature review was made. It is well known that tumors or infection of the adjacent organs, such as the rectum or the sigmoid colon, can invade or infiltrate the bladder. Of these cases, adenocarcinoma of the appendix invading the urinary bladder is very rare and it is very difficult to make a correct diagnosis preoperatively. We describe a case of adenocarcinoma of the appendix who presented with a vesico-appendiceal fistula.th postoperative day. She received adjuvant chemotherapy with 5-fluorouracil and oxalyplatinum. Six years after the operation, the patient remains asymptomatic with no evidence of recurrent or metastatic disease.A 50-year-old woman presented with a 5-month history of urinary frequency and micturition pain. She had no complaints of any intestinal symptoms such as pain, obstruction or melena. She had been treated previously with oral antibiotics for recurrent urinary tract infections. These became more frequent and she started with pain in the lower abdomen and fetid urine. Ultrasonography found a nodular image in the right posterior wall of the bladder. Cystoscopy confirmed the edematous broad-based tumorous lesion in the right posterior wall of the bladder. Transurethral biopsy indicated the presence of a mucinous adenocarcinoma of probable colonic origin. Endoscopy showed a protrusion in the cecum with origin in the appendix and mucoid content. Pelvic enhanced computed tomography (CT) revealed circumferential thickening of the appendix with mucoid material inside and no metastases. The point of the appendix was invading the muscle layer of the bladder, which was thickened . With thet al. gathered a series of 21 cases in Japan [A vesicoenteric fistula is usually a complication of an inflammatory or neoplastic process. Common causes of fistula formation include colonic diverticulitis (51% of the cases), colorectal adenocarcinoma 16%), Crohn's disease 12%) and bladder carcinoma (5%) % and bla. The con%, Crohn'in Japan , as perfin Japan . The preEarly diagnosis of carcinoma of the appendix is difficult because of the absence of specific symptoms. This is because of its anatomical position, which does not interfere with the intestinal passage in its early stage. Therefore, appendiceal carcinoma is always neglected or misdiagnosed. Clinical presentations include acute appendicitis, abdominal mass or intestinal obstruction. In two studies, none of the patients were correctly diagnosed preoperatively ,19 and 4Management of recurrent urinary tract infections should not limit to empiric antibiotic therapy before the exclusion of possible organic causes. Appendiceal carcinoma may invade the bladder without intestinal symptoms but with urinary symptoms only, because of its anatomical position. The recommended treatment for non-carcinoid appendiceal tumours is right hemicolectomy and for T4 tumours en bloc resection of the involved structures. Further study is needed to determine adjuvant therapy.Consent: patient´s written consent was obtained and is available upon request.


The MRI features of epithelioid glioblastoma (eGBM) were analyzed. The apparent diffusion coefficient (ADC), MR perfusion-weighted imaging (PWI), and magnetic resonance spectroscopy (MRS) findings were quantitatively analyzed. The MRI images of 8 cases of eGBM were analyzed retrospectively. The location and edge, signal, peritumoral edema, adjacent meningeal invasion, and enhancement of the lesions were observed. The ADC value, relative cerebral blood volume (rCBV), relative cerebral blood flow (rCBF), and N-acetylaspartate/acetylcholine (NAA/Cho) value were analyzed.n = 3), frontal lobe (n = 3), and parietal lobe (n = 2). The lesion boundary was clear in 6 cases and unclear in 2. The lesions were superficial in 5 cases and in the deep white matter in 3. Internal hemorrhage was observed in 4 cases. There was cystic necrosis in 7 cases, and only 1 case was solid without cystic necrosis. There was no edema around the lesion in 1 case, severe edema in 5, and moderate edema in 2. In 4 cases, the adjacent meninges were involved, and in 1 case, the ependyma was involved. Two patients developed leptomeningeal metastasis within 2 months after the operation. The average ADC value of the tumor parenchyma among all 8 patients was7.15 × 10−4 mm2/s,which was 17.6% lower than that of the contralateral side. The Cho/NAA metabolite ratio was 5.27 and 0.81 in the lesions of 2 patients. The rCBV was 3.51 ml/100 g and 3.32 ml/100 g of lesions in 2 patients; these values were 36% and 29% higher, respectively, than those of the contralateral side. The rCBF was 31.5 ml/100 g/min and 82.1 ml/100 g/min of lesions in two patients; these values were 49% and 203% higher, respectively, than those of the contralateral side. Among the 8 patients, the tumors were mainly located in the temporal lobe ( eGBM characteristics include a superficial location, easy cyst degeneration, easy necrosis and hemorrhage, and clear boundaries. It easily invades adjacent meninges and shows cerebrospinal fluid dissemination and metastasis. Combining new MR techniques, such as ADC values, PWI, and MRS, could be helpful for improving diagnostic accuracy. In 2016, the World Health Organization (WHO) published a new standard for the classification of tumors in the central nervous system[A total of 8 cases of eGBM were collected. Among these, 6 were male and 2 were female patients. The age of the patients ranged from 27 to 68 years old, with an average of 51 years old. The clinical symptoms, treatment, and prognosis are shown in B values were 0 and 1000 s/mm, axial position, slice thickness: 5.5 mm, spacing: 1.0 mm). Enhanced scanning was performed with Gd-DTPA as the contrast medium. Axial, coronal, and sagittal T1WI scans were performed, and the scanning parameters were the same as those used for plain T1WI.A GE3.0T MRI and 32-channel head coil were used. The main scanning sequence and parameters were as follows: T1WI , T2WI , T2 flair , and DWI  and the maximum height of the coronal plane (C). A formula (V = 4/3π × ABC) was used to calculate the volume of tissue including the edema and the tumor (V edema + V tumor) or the tumor volume alone (V tumor). The edema index (EI) was calculated as (V edema + V tumor)/V tumor, with EI = 1 indicating no edema, 1 < EI < 1.5 indicating mild edema, 1.5 ≤ EI < 3 indicating moderate edema, and EI ≥ 3 indicating severe edema [On MRI, the focus location, edge, and internal signal as well as hemorrhage, peritumoral edema, enhancement characteristics, and meningeal metastasis were observed. Brain edema was measured using the following parameters: the maximum diameter of the front and back and the left and right in the axial plane , frontal lobe (n = 3), and parietal lobe (n = 2). The diameters of the lesions ranged from 2.2 to 5.5 cm, with an average of 4 cm. The lesion boundaries were clear in 6 cases and unclear in 2 cases. The lesions were superficial in 5 cases . MRS showed that the NAA peak decreased significantly, while the Cho peak increased accordingly. The Cho/NAA metabolite ratio was 5.27 and 0.81 in 2 patients  that transformed into epithelioid glioblastoma have been reported , suggestMR signal characteristics of the lesions: previous studies have shown that the radiologic characteristics of eGBM are usually similar to those of low-grade gliomas, such as pilocytic astrocytoma, PXA, and ganglioglioma [ioglioma –9. Cystiioglioma , such asioglioma . In thisThe edge of focus and the perifocal edema: the boundaries of most eGBM lesions are clear [re clear . In our re clear . Howeverre clear , 6. In oAdjacent meningeal invasion and cerebrospinal fluid dissemination: in this group of cases, 50% (4/8) of the lesions showed adjacent meningeal invasion and the “meningeal tail sign” after enhancement. Huang et al. [g et al.  reportedg et al. , and thig et al. . Li et ag et al.  reportedThe ADC value can predict the grade of glioma, guide treatment, and predict prognosis . The ADCeGBM should be differentiated from pleomorphic glioblastoma, pleomorphic xanthoastrocytoma, pilocytic astrocytoma, metastatic tumor, etc. Pleomorphic glioblastoma usually occurs in patients aged 45 to 75 years old and is usually located in the deep white matter, while the solid part of the pleomorphic glioblastoma usually shows corolla-like enhancement on contrast-enhanced scans. Pleomorphic xanthoastrocytoma usually occurs in children and adolescents and is especially common in the temporal lobe. Its typical manifestation is cystic degeneration with mural nodules and mild peripheral edema . PilocytThe prognosis of eGBM is very poor. Most of these tumors grow rapidly and invade the adjacent leptomeninges in the early stage. The tumor cells easily spread through the cerebrospinal fluid. Some studies have shown that the incidence of metastasis of eGBM is as high as 69.7% . It alsoThe MRI manifestations of eGBM include some specific characteristics, such as a superficial location, frequent cystic degeneration, necrosis, hemorrhage, a clear boundary, frequent involvement of the adjacent meninges, and frequent spread and metastasis through the cerebrospinal fluid. However, in most cases, it is difficult to distinguish eGBM from other malignant brain tumors. ADC values, PWI, MRS, and new MR technology cannot identify the type of tumor cases, but it is helpful for preoperative tumor grading. Because eGBM can easily invade the adjacent meninges and spread through the cerebrospinal fluid, the prognosis of eGBM is very poor. Its treatment is different from that of ordinary high-grade gliomas. As radiologists, we should carefully analyze each image feature, combining these new MR techniques, to make a more accurate preoperative diagnosis, which can help clinicians choose the best treatment plan.


Intraoperative detection of intrahepatic lesions can be demanding. The use of preoperative contrast-enhanced magnetic resonance imaging (MRI) or computer tomography (CT) combined with intraoperative ultrasound of the liver is state of the art. Near totally regressed colorectal liver metastases (CRLM) after neoadjuvant chemotherapy or nodules in severely altered liver tissue as steatosis or cirrhosis are often hard to detect during the operative procedure. Especially differentiation between benign atypical nodules and malignant tumors can be very difficult. The intraoperative use of contrast-enhanced ultrasound or intraoperative navigation are helpful tools. However, both methods show relevant limitations.The use of intraoperative MRI (ioMRI) can overcome this problem. Relevant structures can be marked within the operative site or immediate control of complete tumor resection can be achieved. This might allow immediate surgical optimization in case of failure.We report the intraoperative application of ioMRI in a case of a 61-year-old male patient suffering from rectal cancer with 10 synchronous bilobar CRLM who was treated stepwise by multimodal treatment and staged hepatectomy. Intraoperative contrast-enhanced MRI of the liver was used during completion procedure of an extended right hemihepatectomy performed as “Associating Liver Partition and Portal vein Ligation for Staged hepatectomy (ALPPS)”.ioMRI provided excellent images and showed absence of liver metastases in the liver remnant. Procedure of ioMRI was safe, fast and feasible.To the best of our knowledge, we describe the first case of intraoperative application of a contrast-enhanced MRI during open liver surgery at the University Hospital of Dresden. Inspection, palpation, and the use of intraoperative ultrasound (IOUS) are important intraoperative tools to detect and localize liver metastases in open liver surgery that are regularly used.However, intraoperative localization and detection of liver tumors can be difficult—especially in patients with severe alteration of the liver tissue such as steatosis, steatohepatitis, or liver cirrhosis. Another challenge for intraoperative localization are small nodules as near totally regressed liver metastases after chemotherapy as well as the differentiation between tumor nodules and scar tissue after resection in patients with repeated liver resections or two-staged hepatectomy.Contrast-enhanced ultrasound, intraoperative use of indocyanine green (ICG) fluorescence imaging, and 3D navigation can help to support the detection of liver tumors. Even though some authors describe the advantages of contrast-enhanced ultrasound of the liver, others show clear limitations of this technique. Intraoperative use of ICG fluorescence imaging in liver surgery can be helpful to detect tumor nodules as well as segment borders within the liver. However, ICG has to be applied a few days before surgery for the detection of tumor nodules .Intraoperative 3D navigation can be helpful, but due to limited experience and variable inaccuracy of this method, the use is actually limited to experimental settings or clinical trials in selected centers.It has been demonstrated that magnetic resonance imaging (MRI) is an excellent imaging modality of the liver. MRI is sometimes the only modality for the detection and differentiation of liver tumors.The first intraoperative magnetic resonance imaging (ioMRI) technique was installed in the 1980s , 3. The The most commonly used application for ioMRI are neurosurgical procedures. However, in recent years, cases and case series of ioMRI for surgery of sarcomas have been published , 8.To the best of our knowledge, this is the first report of an intraoperative use of a MRI during liver resection.We report the case of a 61-year-old male patient suffering from rectal cancer with 10 synchronous bilobar colorectal liver metastases (CRLM) ” procedure with parenchymal transection and additional ligation of the right portal vein. Rubber bands around the right hepatic vein, the right hepatic artery, and the right inflow pedicle were left in situ. Control CT scan on the 7th postoperative day (POD) revealed sufficient hypertrophy of segments II and III. Volumetry of the future liver remnant  showed an increase from initially 440 to 594 cm3 Fig. . HoweverCompletion surgery was performed in the OR-MRI “Combi Suite” as shown in Fig. In our hospital, the so-called “Combi Suite” consists of an MRI  door-to-door to a specific operating room (OR). MRI and OR are equipped with specific connectable tables allowing quick patient transfer from OR table to the ioMRI table by a special transfer board without changing patient’s position.Before using the intraoperative MRI for the patient, cross-training for OR—anesthesiological and MR—personnel has been performed as described by Larson et al. .Furthermore, complete workflow including patient’s transfer from OR to ioMRI as well as the ioMRI was successfully performed with a test person (A.K.). A MRI application specialist guided this procedure.The completion operation was then performed on September 19, 2019.Surgery was performed under general anesthesia. After relaparotomy by reopening a Makuuchi incision, adhesions were cleared and palpation and ultrasound of the liver were performed. Nodules of the right lobe were palpable and visible within the ultrasound. In segment III, the suspicious nodule was then marked by a MRI-specific marker that is normally used for breast-specific MRI diagnostics (Bard® DuaLok ™). Rubber bands around the right hepatic vein, right hepatic artery, and right inflow pedicle were left in place. Surgical instruments and retractors were removed from the patient and the operative table.The abdomen was temporarily closed by positioning an abdominal linen under the abdominal wall covered by a sterile, self-adhesive drape/sheet Fig. .Fig. 5a The patient was then transferred to the ioMRI table by using a special mobile transfer board from the operating table. Both OR and MRI tables were connected and locked and the patient was then transferred to the MRI table on the mobile transfer board Fig. .Anesthesiologic monitoring and equipment were replaced by MRI-compatible devices and disposables. No MRI-compatible equipment, e.g., perfusors, were locked in a MRI-compatible closet while staying connected with the patient Fig. .Anesthesia was performed according to the “practice advisory on anesthetic care for magnetic resonance imaging” of the American Society of Anesthesiologists .The whole team completed a security check before the patient entered the ioMRI room. The phased array body coil was placed around the patient  Fig. .ioMRI of the liver was performed including the application of a liver-specific contrast medium  under continuous standardized cardio-respiratory monitoring. No adverse event occurred during the MRI. Apnea episodes of few seconds were performed for better image quality by the anesthesiologist who was in the MRI room during apnea maneuvers Fig.  g.No heating effect due to abdominal linen was observed.Immediately after completion of ioMRI, the patient was safely transferred back to the OR and relocated to the OR table.The total duration of the MRI was 20 min. The transfer time was round trip 15 min.Intraoperative MRI provided excellent images which demonstrated no suspicious liver lesions in the liver remnant Fig. a and b. Rubber bands were not visible within the MRI images. The breast-specific intrahepatically placed MRI marker was not visible as well. MRI images were immediately analyzed by a radiologist and surgeons.Subsequently, the operative procedure was completed and extended right hemihepatectomy after ALPSS was performed with a total blood loss of 500 ml without any complication.The patient’s postoperative course was regular and the patient left the hospital with normal liver function and closed wounds 10 days after the operative procedure, except a self-limitating bile leakage grade A no morbidity occurred.Improvement of multimodal therapeutic strategies in the treatment of malignant liver tumors as well as the development of surgical techniques and anesthesiological opportunities lead to new aspects in liver surgery.CRLM shows excellent regression under new chemotherapeutic options in some patients. These so-called disappearing liver metastases (DLM) can hardly be detected intraoperatively, especially in patients showing alterations under chemotherapy as chemotherapy-associated steatohepatitis (CASH) or sinusoidal obstruction syndrome (SOS). In addition, repeated liver resections for CRLM with 3 or 4 repeated liver resections are commonly performed in many patients. In those patients, it can be difficult to differentiate between scar tissue and vital tumor in preoperative and intraoperative imaging.Inspection, palpation, and the use of IOUS are standard tools for intraoperative localization of liver metastases in open liver surgery. During minimally invasive liver surgery detection has to be performed by inspection and ultrasound as haptic information is reduced.Intraoperative ultrasound has been described to detect additional lesions not seen in preoperative CT scanning or MRI leading to a change of surgical strategy in 1.4% of cases . The impAnother group favors the use of intraoperative real-time virtual sonography with sonazoid enhancement for the detection of liver metastases < 10 mm .However, some authors describe the superiority of preoperative MRI with higher accuracy over intraoperative contrast-enhanced ultrasound in case of DLM . ShiozawOther authors recommend intraoperative imaging despite good preoperative CT and MRI or a combination of preoperative hepatocyte-specific MRI and intraoperative contrast-enhanced ultrasound for DLM , 16. AnoIn general, we use preoperative MRI if lesions are not visible in CT and/or ultrasound.To our knowledge, there are no data available comparing IOUS with ioMRI of the liver. The main indication for ioMRI is tumors that are difficult to detect in ultrasound.The main entities are hepatocellular carcinomas (HCC) in cirrhotic livers and near-total regressed metastatic lesions under neoadjuvant chemotherapy.We want to clarify that the use of ioMRI is not used instead of ultrasound, but as an additional tool for patients with metastases that were not visible in preoperative ultrasound or for patients with DLM.The use of IOUS remains the standard tool for intraoperative imaging during liver surgery. ioMRI is only used in selected patients.In recent decades, other techniques for intraoperative detection of hepatic lesions were implemented and need to be shortly discussed. There are different methods of intraoperative image guidance as to the application of ICG or intraoperative real-time 3D navigation which can be supportive in detecting intrahepatic lesions. The use of ICG has been shown to detect HCCs as well as CRLM if ICG was applied several days before surgery. However, this method has clear limitations in the detection of small lesions like DLM, especially if located > 5–10 mm distant to the liver surface [Intraoperative 3D navigation is a promising approach—especially for DLM. But due to a lack of accuracy after intraoperative liver shifting this approach is not yet relevant in routine use.Another more invasive method to detect DLM is the interventional placement of fiducials in each  metastasis before starting the neoadjuvant chemotherapy to clearly identify the lesions even after macroscopically complete regression .After the first description of interventional and intraoperative MRI suites in the 1980s, the ioMRI technique was mainly applied in neurosurgical procedures , 19.Technical as well as neurosurgical improvement of this technique over the last 30 years led to higher safety and accuracy. Formation of expert and consensus groups as the German Study Group for Intraoperative Magnetic Resonance Imaging or recommendations on anesthetic care for magnetic resonance imaging by the American Society of Anesthesiologists has been elaborated .The general experience and technical developments within the use of intraoperative MRI made this procedure safe and feasible—even in abdominal surgery.To our knowledge, the intraoperative use of MRI of the liver has not been described before. There are no preceding reports or experiences regarding this technique. No information regarding disturbances of images by open abdomen or intraabdominal material and no information about heating effects on the open abdomen are available.We could show that ioMRI is safe and feasible even in the patient with a provisory-closed abdomen during liver surgery.In our opinion, safety is depending on the technical setting. A door-to-door connection between OR and MRI (as seen in our Dresden Combi Suite) is essential for an acceptable time frame and circumstances of sterility.Careful consideration of high-risk patients is of utmost importance. High-risk patients including patients with critical NYHA state do not qualify for this procedure as well as patients with pacemaker and defibrillator (ICD).One unknown factor was the temporary closure of the abdominal wall as wet tissues are at risk to develop heating effects and possible disturbances of the MRI images. We showed that temporary closure of the abdominal wall by abdominal linen and self-adhesive sheet was safe without heating or burning problems. Interestingly, no disturbances of the MRI images were seen due to the tissue or the rubber bands. However, the use of a small marker is not sufficient for liver images. Other markers, e.g., liquid-containing markers, need to be tested in the future. Images of the ioMRI were of high quality and influenced further surgical procedure: in our patient, additional liver resection was avoided by the use of the ioMRI.It has to be considered that there are some limitations for ioMRI during liver surgery:Patient-specific limitations for ioMRI during liver surgery are MRI-incompatible implants such as pacemaker and ICD and any other metallic foreign body. Despite the fact that there is no weight limit for the ioMRI used in our hospital, the body size and girth/circumference of patient’s body is an important limitation. The patient has to fit in the bore. In the ioMRI used in this case, the maximum diameter of the bore is 70 cm.Besides patient-specific limitations, major concerns are the additional costs and limited availability of the ioMRI.The costs of the ioMRI-imaging are about 750, − euros/h including the imaging and radiological personnel. In this case, the additional costs are calculated with about 350, − euros. Noteworthy, as the Combi Suite—MRI is also used for non-intraoperative MRI, the costs of a regular MRI and the ioMRI are identical. There are no additional costs for patient transport as the ioMRI and the OR are designed as Combi Suite and both rooms are connected door-to-door. Additional costs for the occupation of OR and surgical and anesthesiological personnel during ioMRI-procedure cannot be provided in a valid calculation.To our knowledge, we describe the first intraoperative MRI during an open liver resection. The use of ioMRI of the liver is safe and feasible and provides excellent images of the liver.The application of intraoperative liver MRI is not necessary for routine hepatectomies but can provide excellent information in selected patients. In our opinion, the use of intraoperative liver MRI is promising in detecting sonographic occult liver tumors. Moreover, documentation of complete resection of hepatic tumors immediately after resection can avoid R1-/R2-situations and early reoperation and should reduce the rate of intrahepatic tumor recurrence. However, this needs to be proven in further studies.


We report two patients who presented with small bowel obstruction secondary to gallstones in the ileum. Both patients were geriatric women with multiple comorbidities. The first patient was a 73-year-old woman who presented with a gallstone eroding and obstructing the duodenum (Bouveret's syndrome) secondary to gallbladder cancer with diffuse metastatic spread to the liver. The stone was disimpacted endoscopically using lithotripsy. The patient presented two days later after the stone had migrated downstream into the small bowel causing obstruction requiring surgical intervention. Second patient was an 81-year-old woman who presented with small bowel obstruction caused by a gallbladder stone impacted in the distal ileum. Both patients were managed laparoscopically with a mini laparotomy to extract the affected segment of bowel loop via small incision on the anterior abdominal wall at the port site with enterolithotomy. Both patients were discharged by postoperative day four with no complications. We conclude that, in elderly patients with multiple comorbidities presenting with gallstone ileus, laparoscopic approach provides early recovery with minimal pain. Gallstone ileus is a rare complication of cholelithiasis which is defined as a mechanical intestinal obstruction caused by impaction of one or more gallstones in the ileum after being passed through a biliary-enteric fistula . Its manFirst case is a 73-year-old, weak, and debilitated woman with a Karnofsky index of 40  who was known to have gallbladder cancer with multiple metastatic nodules to liver, anemia, prior stroke with residual weakness, and hypothyroidism. She presented to the emergency room with nausea, vomiting, and abdominal discomfort. Computed tomography (CT) imaging revealed impacted stone in the duodenum causing gastric outlet obstruction Figure . She undAn 81-year-old female with a prior history of unresectable lung cancer status post radiation therapy, dementia, and multiple other comorbidities presented to the emergency room with acute onset nausea, vomiting, abdominal discomfort, and bloating. She was afebrile with vital signs significant for a blood pressure of 100/50 mmHg. Physical examination was significant for mild abdominal distention. Laboratory results were significant for AKI with a BUN of 81 mg/dL and a creatinine of 4.2 mg/dL (baseline 1.5 mg/dL). The patient underwent a CT scan of the abdomen that revealed pneumobilia and small bowel obstruction secondary to a radiolucent impacted gallstone in the distal ileum Figure . An enteThe classic clinical presentation of gallstone ileus in an older woman with episodic subacute obstruction with nausea, vomiting, crampy abdominal pain, and/or variable distension is commonly present as was seen in both patients before surgical intervention . The epiComputed tomography (CT) is the preferred modality in comparison to plain abdominal films or ultrasound in the diagnosis of gallstone ileus with a sensitivity of up to 93% . When diMultiple surgical approaches to relieve the obstruction have been defined in the literature, but, since this condition is so unusual, there has been no accepted consensus regarding its management. Our approach was a mini-laparotomy at the supraumbilical port for stone extraction, however, the current surgical approaches include: simple enterolithotomy as was performed; enterolithotomy, cholecystectomy and fistula closure (one-stage procedure); and enterolithotomy with cholecystectomy performed later (two-stage procedure) . EnterolGallstone ileus is primarily a geriatric disease in patients with multiple comorbidities. Use of a laparoscope and a mini laparotomy to perform an enterolithotomy provides short operative time with minimal postoperative morbidity and early recovery.


Pleomorphic adenoma (PA) mostly involves parotid glands, while extra-parotid localizations are relatively uncommon. Particularly, PAs of the cheek minor salivary glands with a size larger than 4 cm are exceedingly rare, with only few cases reported. Surgical treatment of PA usually consists in radical excision. However, despite a presumptive radicality, recurrences, sometimes followed by malignant transformation, may occur. Here we report a case of a large-sized (6 cm) PA of the cheek minor salivary glands in a 70 year-old female patient, successfully treated through a conservative approach, based on the use of Nd:YAG Laser (λ=1064 nm). No recurrences were observed after a 2-year follow-up. A concise review of the literature, describing the features of 14 cases is also provided. Advantages of laser treatment include a precise cut, reduction of trauma on surrounding tissues, the possibility of a very good intraoperative hemostasis. Such features may sometimes allow to avoid general anesthesia, even for removal of big lesions. Post-operative course, in terms of pain and swelling, is usually better for intervention performed with laser, when compared to traditional surgery. Key words:Oral surgery, oral pathology, pleomorphic adenoma, laser surgery, minor salivary glands, salivary glands tumors. Pleomorphic adenoma (PA) is the most common benign neoplasm of salivary glands, accounting approximately for 70% of all salivary neoplasms. PA occurs between the 3rd and the 6th decade of life with a slight female predilection. Only 8% of PAs affect minor salivary glands, the most common intra-oral sites being the palate (60-65%), followed by the lip 10%) and buccal mucosa. PAs affecting minor salivary glands of the cheek are very rare, accounting for less than 1% of all PAs 0% and bu. Here weA 70-year-old female patient was referred for a painless, slow-growing swelling within the right cheek, which was present since the last 15 years and significantly increased during the last year. Extraoral clinical examination revealed asymmetry of the face caused by the presence of a mobile, hard-elastic mass. Intraoral evaluation showed a swelling covered by intact mucosa, with a dimension on the major size of approximately 5 cm. At magnetic resonance imaging (MRI), the mass showed well-defined margins (most likely associated to the presence of a capsule), smooth borders, inhomogeneous aspect and a high-intensity T2-weighted signal. The mass was adherent to Bichat’s fat pad, delimited by the Stensen’s duct and masseter muscle, the upper fornix and lower fornix posteriorly and the facial artery anteriorly  was performed under local anesthesia  and/or computed tomography (CT) prior to MRI, we performed MRI ,2,4,14. et al. reported a peculiar case of PA showing bone and bone marrow formation within chondroid tissue (et al. reported a case of perineural involvement of a small nerve associated with a benign PA (From the pathologic macroscopic point of view, PAs of minor salivary glands are often grossly encapsulated and they usually range from 1 to 4 cm in size . Larger d tissue . In the enign PA .PAs might sometimes recur, either due to seeding or inadequate removal ,12. PA dMoreover, PAs show sometimes signs of extracapsular spread at postoperative histopathologic examination . Such a In our case, Nd:YAG laser was chosen for its precise and minimally invasive cut, which allows radical excision in most cases. Nd:YAG laser wavelength also provides a powerful haemostatic activity .The energy transferred by the laser beam to biological tissues results in tissue heating. The mean thickness of epithelial, stromal and vascular changes induced by Nd:YAG laser, as detected at microscopic evaluation, is 363.7, 459.1 and 169.85 micron, respectively . It has Nd:YAG laser also shows a strong antimicrobial activity, which reduces the need for antibiotics and the rate of infectious complication, even in cases of second intention wound healing . In the


The pyriform sinus fistula is a rare condition described as an epithelialized tract connecting the skin of the neck to the foregut, and may result in cervical cysts and iterative abscesses misleading the diagnosis.The clinical and radiological examinations are all useful. Surgery stands as one of the most effective therapeutic options consisting on the total excision on the eventual cyst, and the fistula that is followed to its inner opening on the pyriform sinus.We present a case of a 3-years-old boy with a pyriform sinus fistula that caused recurrent neck abscesses treated independently delaying the diagnosis. Once in our structure, after radiological examination and antibiotics to cool the infection down, the surgery removed the cyst with its tract that opened in the pyriform sinus. The follow up showed an effective result with the total disappearance of the lesion with no more infectious episodes.Even if it's a rare condition, the diagnosis of apyriform sinus fistula must be considered in front of every patient with a history of recurrentlatero cervical abscess. •The pyriform sinus fistula is a rare condition described as an epithelialized tract connecting the skin of the neck to the foregut, and may result in cervical cysts and iterative abscesses misleading the diagnosis•The clinical and radiological examinations are all useful•Surgery stands as one of the most effective therapeutic options consisting on the total excision on the eventual cyst, and the fistula that is followed to its inner opening on the pyriform sinus Any improvement was observed besides the disappearance of the infectious signs. And, for the persistence of the lesion, and the recurrence of the abscesses, the patient ended up consulting in our structure.The clinical examination found a left cervical soft cystic lesion, over the anterior border of the sternocleidomastoid muscle, with inflammatory tissues around, fistulated to the skin. With no other abnormality, normal tonsil lodges and no lymphadenopathies.On the computed tomography (CT) imaging, the lesion appeared as a thickening of the left cervical soft tissues. No cystic aspect nor septic collection nor air within the thyroid gland was identified .Fig. 1CTThe patient was thus, programmed for an exploratory cervicotomy after seven days of antibiotics to cool the infection down, until the disappearance of the inflammatory skin signs.The surgery was performed under general anaesthesia, by a senior resident with 4 years of specialised training and consisted on an excision of the lesion following its tract, which passed posterior to the carotid artery, until the superior part of the thyroid gland, which appeared not to be affected and therefore was left intact as well as the recurrent laryngeal nerve and the superior laryngeal nerve. The thyroid cartilage and the inferior pharyngeal constrictor muscle were also spared. No anatomical relation was found between the tract and the hypoglossal nerve. And its trajectory never descended into the mediastinum. Finally, its sinus opening within the pyriform fossa was visualized . No direThe histological analysis of the operatory piece didn't find any skin appendagesnor cartilaginous residue confirming the ectodermic origin.The follow up, on 8 months until today, showed a total disappearance of the cystic lesion with no more infectious episodes nor cervical abscesses.This case has been reported in line with the SCARE 2018 criteria .33.1The second, third and fourthbranchial ectodermic clefts form the cervical sinus of His which opens into the skin through one single orifice overlying the anterior border of the sternomastoid muscle, which is the location of the embryologic cervical sinus ,5. Thus,The classic description of thepathogenesisis based on the persistence of the pharyngobranchial duct, which connects the third and fourth pharyngeal pouches to the pharynx and normally degenerates during the seventh week of development, resulting in a sinus tract that communicates with the pyriform fossa .3.2The course of the fistulae is located under the branchial derivatives of the associated arch and above the derivatives of the following one .The thirdbranchial cleft fistula arises from the rostral end of the pyriform fossa  and woulThe fourthbranchialcleft fistula arises, lower than the third, from the pyriform sinus apex andfollows a more indirect route, from the pyriform fossa, medial and below to the superior laryngeal nerve, above the carotid bifurcation . It then3.3However, some clinical elements seem not to match with the previous established pathogenesis based on the direct communication across the branchial arch from pouch to cleft . Indeed,To better fit the clinical presentation of these anomalies, James et al. proposed that theses fistulae derive from the thymopharyngeal duct, formed while the thymus descends during the seventh and eighth weeks, like the cysts formed along the thyroglossal tract while the thyroid gland descends from the tongue base to its final site. Besides, the simultaneity of these two events may explain that a failure of the thymopharyngeal duct to close would lead to the formation of a branchial sinus lined with endodermal cells arising from the piriform fossa and passing, in close association with the thyroid, toward the cervical inlet .3.4The pyriform sinus fistulae are an extremely rare condition ,8. Descr3.5The clinical aspect varies with age. In the neonate, the condition is dangerous as the fistula can rapidly get enlarged with the infant swallowing saliva or milk, leading to tracheal compression, a stridor and respiratory distress .In an older patient, the typical form ismore likely to appear as a laterocervical swelling, mostly described on the left side. Embryologically, the left fourth branchial arch artery forms part of the aortic arch, while the right one becomes the right subclavian artery. This normal asymmetry may be related to the noted left-sided predominance of the fistula, so the left-sided predominance may correspond to the fourth pouch origin of this condition .The lesion is frequently exposed to infection causing recurrent lateral neck abscesses or cellulitis, as in our case and up to 42% according to literature , which iThe combination of a congenital sinus and acute suppurative thyroiditis was first described in the Japanese literature. Then, several similar cases have been reported subsequently, which are now believed to be infections arising from an underlying congenital pyriform sinus fistula, probably a remnant of the third or fourth branchial pouch .In another hand, the noncommunicating or non infected communicating cysts may present as cold thyroid nodules. And the cysts that are partly or completely intrathyroidcan be confused with thyroglossal duct cysts .Also, thOther signs can possibly be associated such as dysphagia, odynophagia, sore throat, hoarseness, limitation of cervical extension, cough, upper respiratory infections, and even mediastinitisin a context of fever ,7.3.6According to the most common clinical presentation, the CT or magnetic resonance (MR) imaging is often the radiologic study of choice in delineating inflammatory lesions along the tract, even though the sinus or fistulous tract was not clinically apparent. With a superiority of the contrast-enhanced CT in detecting air density at the fistulous tract as well as in depicting thyroid gland involvement as a loss of high density in the affected gland, with a higher resolution .The CT may reveal abnormal soft tissue swelling, an enhancing, often cystic mass associated with fat streaking and hypodensity within the ipsilateral  thyroid lobe ,3.The paUltrasonography (US), even if limited in its ability to depict the hypopharyngeal lesions , can alsBarium esophagography can also be used to expose the hypopharyngeal fistulous opening. Its sensitivity varies, up to 50%  or even It is also interesting to observe the fistulous opening in the pyriform sinus through nasal fiberoptic endoscopy or through rigid pharyngoscopy or a direct laryngoscopy at the time of the surgery, even during acute episodes . When th3.7The patient care should start by the administration of appropriate antibiotics to decrease the surrounding inflammation .The surgical excision is the definitive treatment, performed several weeks after the infection has been resolved . It is bThe actual standard should add a hemithyroidectomy, if the gland is clearly affected. The main risk remains the recurrent laryngeal nerve, intimately associated with the tract, and often surrounded by fibrotic tissue resulting from chronic thyroiditis and recurrent abscesses . AttentiTo ease the localisation on the sinus opening, an internal or external catheterism of the fistula can be done with a Fogarty catheter .Is also Chemocauterization or electrocauterization are less invasive, but less efficient alternatives with a higher rate of recurrence. The chemocauterization with 40% trichloroacetic acid (TCA) with or without primary mucosal coverage allows the obliteration of a longer segment of the fistulous tract depending on the deep of the acid penetration, with less risk to adjacent structures from heat and electrical spread and it can be safely repeated .4The pyriform sinus fistula is a rare condition but must be considered in front of every patient with a history of recurrent cervical abscess, in either side of the neck, in any child or young adult.Also, it has become accepted that thyroid abscesses in children often indicate an underlying branchial remnant, especially when cultures reveal a mixed flora ,2.The surgical treatment appears to be the most effective in order to cure the condition, with a satisfying postoperative follow-up.We presented a case report that supports the literature data concerning the clinical and therapeutic aspects of the pyriform sinus fistula.This work has been approved by the ethical committee of our department.The parents of the young patient (3 years old) gave their written informed consent for the surgery and the follow up.This research did not receive any grant or funding from governmental or private sectors.Rabii Laababsi: Corresponding author writing the paper.Khadija El Bouhmadi: Writing the paper.Sami Rouadi: Study concept.Redallah Larbi Abada: Study concept.Mohamed Roubal: Correction of the paper.Mohamed Mahtar: Correction of the paper.Name of the registry: Research registry.Unique identifying number or registration ID: 5848.https://www.researchregistry.com/browse-the-registry#home/registrationdetails/5f2028117a1cfd0016771167/Hyperlink to your specific registration (must be publicly accessible and will be checked): Rabii Laababsi.The patient's parents gave informed consent for publication.Not commissioned, externally peer reviewed.All authors have no conflict of interest or financial support with this article.