This case report describes for the first time the cytologic characteristics of a hormonally secreting pituitary adenoma in a cat. An 8-year-old female spayed domestic long-haired cat was referred with a previous diagnosis of hypersomatotropism and secondary diabetes mellitus 7 months prior. Clinical signs included weight loss, polyphagia, polyuria, and polydipsia. Serum insulin-like growth factor-1 was 340 nmol/L (RI: 12-92), and CT scan revealed a hypophyseal mass, and a presumptive diagnosis of acromegaly was made. A transsphenoidal hypophysectomy was performed. A fragment of the pituitary gland was subjected to a squash preparation and cytology revealed a neuroendocrine neoplasm characterized by anisokaryosis and prominent nucleoli. Additional cytologic findings included cell cohesiveness, indistinct cytoplasmic borders, nuclear crowding, molding, and fragmentation. A diagnosis of adenoma was based on a lack of histopathologic or imaging evidence of invasion. A week later, during post-surgical hospitalization, the patient worsened and died. Histopathology from a necropsy procedure revealed fibrinosuppurative meningitis as a post-surgical complication. Pituitary adenomas might have an aggressive cytologic appearance, despite a lack of histopathologic invasion or dissemination. The prevalence of pituitary adenomas varies from 15 to 20% in people –5. TheseAnatomic and histopathologic findings have been extensively described in animals, but scientific reports approaching the cytologic features of pituitary neoplasms are still lacking. The objective of this report is to describe the findings of an intraoperative imprint smear cytology from a somatotroph pituitary adenoma in a cat, in the context of a complete diagnostic work-up protocol. This case report was written in agreement with the most updated version of the CARE guidelines .An 8-year-old female spayed domestic long-haired cat was referred to the neurology and neurosurgery service of the UC Davis William Pritchard Veterinary Medical Teaching Hospital for transsphenoidal hypophysectomy. The patient received a diagnosis of hypersomatotropism  and secondary diabetes mellitus 7 months prior to admission. Insulin therapy had been discontinued for about 6 months, and since then, the patient had lost ~680 g in weight and developed polyuria and polydipsia. On physical examination, the patient weighed 3 kg (body condition score = 3/9), and was bright, alert, responsive, and hydrated. Prognathism and prominent facial features were noted. No apparent cardiovascular, respiratory, integumentary, gastrointestinal, or neurologic abnormalities were noted, and no relevant familial history was reported.Prior to the surgical procedure, a complete blood count  revealed moderate lymphopenia , and slight thrombocytosis . The biochemical panel  revealed mixed metabolic acidosis and alkalosis , selective chloride loss , mild hyperkalemia , marked hyperglycemia  and mild hypertriglyceridemia . Serum fructosamine levels were 683 μmol/L (RI = 190–365) and a thyroid panel  revealed total T4 < 0.5 μg/dL (RI = 1.1–3.3), free T4 <0.3 ng/dL (RI = 0.59–3.04) and TSH <0.03 (RI = 0-0.07). Urinalysis was performed on a sample obtained by cystocentesis, which revealed marked glucosuria, mild proteinuria and hypersthenuria .Pre-surgical transverse contrast-enhanced CT images of the brain revealed a hyperintense large ovoid-shaped mass in the area of the pituitary gland measuring 8.6 by 8.3 by 6.8 mm .During the transsphenoidal hypophysectomy, imprint smears from the pituitary mass were prepared and submitted for cytologic analysis. The specimen was highly cellular, and the background contained a diffuse and amorphous light pink material (consistent with fragmented cytoplasms) and a low number of red blood cells, admixed with many naked nuclei and other cellular debris. Intact nucleated cells were scattered throughout in vaguely cohesive sheets and clusters and presented indistinct borders, intermediate nucleus:cytoplasm (N:C) ratio, and a pale blue to pink smooth cytoplasm. The nuclei of this population of cells were oval to polygonal with finely granular chromatin patterns and multiple small and prominent nucleoli. Anisokaryosis and anisocytosis were marked. Nuclear crowding, molding, and fragmentation were noted. Rare endothelial cells were also noted. Findings were interpreted as neuroendocrine neoplasia .On histopathologic evaluation, the pituitary gland tissue consisted of a densely cellular, expansile, unencapsulated mass composed of nests and cords of polygonal cells on a fine fibrovascular stroma. Neoplastic cells presented distinct cell borders and a moderate amount of brightly eosinophilic glassy to granular cytoplasm. Nuclei were round with vesiculated chromatin and one prominent nucleolus. Anisocytosis and anisokaryosis were mild. Mitotic figures were rare . The perHistochemical staining with reticulin and periodic acid Schiff (PAS) and immunohistochemical staining with growth hormone were performed. Reticulin staining revealed a loss of reticular network within the neoplasm with respect to the compressed peripheral, non-neoplastic tissue , and theA week later, during post-surgical hospitalization, the patient's clinical condition worsened, and it died. A post-mortem examination with histopathology revealed fibrinosuppurative meningitis as a post-surgical complication, which was likely responsible for this patient's death. Other findings included a small focal area of thyroid nodular hyperplasia, vasculitis with thrombi in the lungs, multivacuolar degeneration, associated with focal amyloidosis and atrophy of the endocrine pancreatic islets, focally extensive lymphocytic enteritis, an incidental focal dilation in the esophagus and moderate, and diffuse vacuolar degeneration of the liver. Evaluation of remnant pituitary tissue in the sella turcica, as well as regional lymph nodes, confirmed a lack of invasion into the surrounding vasculature, parenchyma, bone, or lymphatics.This is, to the best of the author's knowledge, the first cytologic description of a pituitary adenoma in animals. The cytologic appearance of the tumor exhibited some malignancy criteria such as marked anisokaryosis, prominent nucleoli, and nuclear molding. Despite these features, histological evaluation, in conjunction with CT and postmortem findings, was consistent with the accepted definition of an adenoma , 12. Celvia cytology was achieved in over 90% of human, canine, and feline cases in studies investigating over 4,000 correlated biopsies  secretion by another pituitary gland impaired function as an additional contributor to the polyuria could be speculated.One potential limitation of this report might be represented by the lack of cerebrospinal fluid (CSF) analysis done prior to surgery. As stated earlier, according to a recent WHO consensus, cerebrospinal involvement is used in the assessment of pituitary gland malignancy . HoweverPituitary adenomas in cats may have a relatively aggressive cytologic picture and care should be taken not to presume malignancy. If tumor growth is causing secondary pituitary atrophy, resulting central endocrinopathies, such as hypothyroidism, should be ruled out.The original contributions presented in the study are included in the article/supplementary material, further inquiries can be directed to the corresponding author/s.FA and KN contributed to conception and design of the case description. FA organized the case data and wrote the first draft of the manuscript. KN and MC wrote sections of the manuscript. All authors contributed to manuscript revision, read, 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.All claims expressed in this article are solely those of the authors and do not necessarily represent those of their affiliated organizations, or those of the publisher, the editors and the reviewers. Any product that may be evaluated in this article, or claim that may be made by its manufacturer, is not guaranteed or endorsed by the publisher.


The authors present a case of cholesteatoma of external auditory canal (CEAC) with extensive invasion of mastoid; ossicle chain and tympanic membrane remained intact. The only symptom was chronic otorrhea. Diagnosis was based on clinical elements and CT scan was used to measure pathology and program surgery. Treatment was modified radical mastoidectomy associated with meatoplasty. Due to the insidious character of CEAC and the proximity with important structures of the external auditory canal, it must be always considered in differential diagnosis for lesions of external auditory canal. This case report intended to review clinical and surgical aspects of treatment of CEAC and present our approach in a case with severe lesions. Cholesteatoma of the external auditory canal (CEAC) is a rare condition affecting especially elderly people. The progress of the disease is slow and the symptoms are not too evident. This leads to late diagnosis, with progressive bone lysis affecting important circumjacent structuresThe authors present a case of CEAC with extensive invasion of the mastoid, exposing the lateral sinus and the dura. Hearing acuity and structures of the tympanic cavity were unaffected.We discuss the possible causes and treatment of choice.In clinical practice of ear diseases, cholesteatoma occurs on the tympanum-mastoid segment. It rarely originates from the external auditory canal (EAC), whose incidence is about 0.1-0.5% in new patients with ear problemsIn 1850In CEAC, lateral epithelium desquamation is affected. Keratin debris are blocked, causing local erosion and bone lysis, which can be severeDiagnosis is made through physical examination. There is otalgia, otorrhea, hearing acuity is unaffected; otoscopy shows an intact tympanic membrane and erosion on a distinct spot of the external auditory canalDifferential diagnosis for CEAC should include necrotizing external otitis, tumors and KO. The latter is more frequent in young adults presenting severe otalgia and bilateral sensorial hearing loss. Differently from CEAC, which usually affects older patients, KO is unilateral, there is otorrhea, chronic pain and hearing acuity is preserved. In KO, the EAC has a keratin plug; otoscopy, following keratin plug removal, shows stenosis, redness and granulation tissue. In CEAC, there is an epidermal diverticulum on the lowest part of the canal, with epidermal debris and otorrhea; the other parts of the EAC are normalThe treatment of CEAC may be clinical or surgical. The former is carried out through local flushing and topic antibiotics. The latter is through resection of the cholesteatoma and necrotic boneClinical treatment is indicated whenever the lesion is limited and there is no otalgia, in cases that surgery is contraindicated, in patients refusing surgical procedures and, when observation without treatment is an option to evaluate progressionSurgical treatment is indicated in the following situations: chronic pain (despite treatment), frequent infection , facial palsy or chronic dizziness, progression during follow-up, involvement of hypotympanum, jugular or mastoid (showed in CT scans), diabetes mellitus or immunosuppression Patient A. F., male, 59 years old. Visited the otorhinolaryngological outpatient facility showing otorrhea, otalgia and ear fullness on the right for ten (10) years. Otoscopy on his right ear showed epidermal debris in the canal. Following debris removal, the tympanic membrane was intact, and there was severe erosion of the posteroinferior quadrant of the EAC. Otoscopy of the left ear was normal. Bilateral pure tone audiometry was normal.We suspected of CEAC and requested a CT scan of temporal bones. CT scan showed erosion of the posterior wall of the EAC, mastoid involvement and exposure of the lateral sinus and meninges .Figure 1Transoperative observation revealed erosion of the entire posterior wall of the EAC, except for a thin bone band close to the tympanic ring . The tymWe conducted modified radical mastoidectomy type I plus meatoplasty. The patient showed good outcomes and normal hearing, without recurrences, in a postoperative period of two years.CEAC, due to its slow and insidious progression, may severely affect structures of the middle ear at the time of presentation. In our case report, the lesion was severe at first presentation, and we were not able to visualize its limits through otoscopy. CT studies were important to determine the severity of the disease and involvement of other structures; therefore, CT scan is indicated whenever CEAC is suspected.The disease had possibly appeared spontaneously, since the patient did not have any predisposing factor to develop CEAC.Mastoid invasion is one of the indications for surgery. After the patient agreed to undergo surgery, we performed modified radical mastoidectomy and wide meatoplasty; thus, we could achieve good control of the cavity during the postoperative period.We were able to preserve the tympanic cavity and its structures; therefore, the surgical procedure was able to cure and preserve the patient's hearing acuity. It is worth mentioning that, in spite of the increased progression of the disease, there was no involvement of the facial nerve, inner ear or nerves emerging from the jugular foramen.Otorhinolaryngologists must suspect of CEAC in cases of chronic otorrhea and intact tympanic membrane. After diagnosis of CEAC, CT scans of the temporal bone must be performed in all patients to evaluate the real severity of the disease and involvement of circumjacent structures . In most cases, the treatment of choice is surgery, whose main purpose is to eradicate the lesion and, if possible, preserve the patient's hearing acuity. The modified radical mastoidectomy is the most common surgical procedure carried out in these cases.


Sessile serrated lesions (SSLs) are serrated polyps (SP) with the typical serrated architecture of the crypt lining epithelium. SSL has an important clinical implication because they are recognized as precursor lesion of sporadic colorectal cancer (CRC) through “serrated pathway.” SSLs usually appear flat to sessile, and are located in the right colon.A 69-year-old man was referred to a tertiary medical center because of intermittent hematochezia for 2 years.Colonoscopy revealed a large, pedunculated polyp in the rectum. The polyp surface was slightly reddish in color and the elongated stalk was covered with almost normal mucosa. Histopathological examination of the resected specimens revealed the typical features of SSL with low-grade dysplasia.Endoscopic mucosal resection using a detachable snare was performed on the tumor for definite diagnosis and treatment.There was no evidence of immediate or delayed bleeding after endoscopic mucosal resection, and the hemoglobin level normalized after a 1-year follow-up.We report a rare case of a large pedunculated polyp with typical histological features of SSLs in the rectum. Endoscopists should always consider SSLs at any location even with unusual morphological findings. Recently, the sessile serrated lesion (SSL) is a new terminology that replace SSA/P according to the 2019 World Health Organization (WHO) classification. SSLs are clinically significant because they are potential precursors of sporadic microsatellite unstable colorectal cancer (CRC), accounting for 15% to 30% of all CRCs.,4 Somatic BRAF mutations and DNA CpG island hypermethylation (CIMP) are the key molecular features of the serrated pathway.,6In 1990, Longacre and Fenoglio-Preiser first described polyps characterized by a serrated architecture and unequivocal dysplasia known as sessile serrated adenoma/polyp (SSA/P). These lesions are predominantly found in the proximal colon. As rare forms of SSLs, pedunculated polyps or depressed surfaces have also been described.,10 Most SSLs with these features were also located in the right-sided colon and small in size. Herein, we report a large pedunculated polyp with the typical histological features of SSLs in the rectum.Morphologically, SSLs are usually flat to sessile and similar in color to the surrounding mucosa. Under endoscopy, the presence of a mucus cap, rim of bubbles or debris, alteration of the contour of the mucosal fold, and/or loss of the normal mucosal vascular pattern also suggest the possibility of SSLs.A 69-year-old man was referred to our clinic with intermittent, scanty hematochezia and dizziness that had persisted for 2 years. He had no medical history and had never undergone a colonoscopy. The patient complained of a mass in his anus that had spontaneously reduced. No signs of hemodynamic instability were observed during outpatient clinic visits. The vital signs of the patient were as follows: blood pressure, 122/53 mm Hg; pulse rate, 82 beats per minute; and body temperature, 36.9°C. A physical examination revealed no specific findings. The initial laboratory data showed a hemoglobin concentration of 3.7 g/dL .Initially, colonoscopy was performed to identify the cause of anemia and hematochezia. Colonoscopy revealed a large, pedunculated polyp in the rectum. However, we could not find any colorectal polyps other than rectal lesions during initial colonoscopy. The surface of the polyp was slightly reddish in color and was surrounded by mucus without a peripheral rim of debris or bubbles. The elongated stalk was covered with almost normal mucosa Fig. . A biops And no histological evidence of cytoplasmic eosinophilia or ectopic crypt formation was observed. Thus, we were able to differentiate the lesion from the TSA. The stalk of the polyp consisted of loose connective tissue within the submucosal layer  up to the year follow-up.The requirement for institutional review board approval was waived owing to the retrospective nature of the study. Written informed consent was obtained from the patient and his legal guardian for the publication of this article and any accompanying images. Serrated lesions with abnormal proliferation are characterized by horizontally growing crypts along the muscularis mucosa, a dilated crypt base, serrated morphology throughout the crypt, including the base, and asymmetrical proliferative features of the crypt. This is caused by the migration of the proliferative zone to the side of the crypt, which is a distinguishing feature of HPs. SSLs are clinically important because, as recent research has indicated, they are the precursor lesions of the serrated pathway to CRC.The 2019 WHO recognized 4 categories of serrated colorectal lesions: HP, SSL, SSLs with dysplasia, traditional serrated adenomas, and unclassified adenomas. Kim et al reported a pedunculated polyp with histopathological features of SSLs, and all pedunculated polyps showed a BRAF-V600E mutation. Previous studies have suggested the possibility of a relationship between BRAF mutations and apoptosis; thus, it may play an important role in the developmental process of flat and depressed morphology. In this case, wild-type BRAF may be related to pedunculated morphology, although this has not yet been well established. Furthermore, it is possible that the lesion is KRAS-associated serrated, although KRAS mutations have been identified in approximately 6% of Asian patients. Unfortunately, we were unable to confirm the KRAS mutation in this case.BRAF is a member of the RAF family of serine/threonine-protein kinases. As a result of the V600E mutation, BRAF and its pathway are activated and are strongly associated with CIMP-positive cancers. BRAF mutations are reported to be present in 78% to 100% of SSLs and 0% to 0.4% of tubular adenomas, supporting the importance of SSLs as precursors of CRC.,15,16 However, more recent study presented high prevalence (up to 15%) of SSA/Ps in a screening population by high-detecting endoscopist and an experienced pathologist. SSLs usually appear flat or sessile endoscopically; 98.1% of lesions are flat according to the Paris classification, and 75% to 90% are right-sided. In addition, they have soft and smooth surfaces and are often covered with mucus caps. However, the morphological characteristics of SSLs remain poorly characterized. Recent studies have suggested an unusual form of SSLs within the spectrum of serrated neoplasia.,10 Kim et al described 5 pedunculated SP with histological features of SSLs. These polyps showed a pedunculated configuration consisting of a small globular head and short and weak pedicle. All polyps were proximally located and were less than 1 cm in size. Another study described SSLs with a depressed surface, which, according to the Paris classification, corresponds to type 0-IIa + IIc or 0-Is + IIc. Colonic polyps rarely cause symptoms and are usually incidentally detected. Furthermore, previous studies have suggested that SSLs may cause fewer symptoms than adenomas, making them difficult to detect clinically. Despite a pathologically confirmed SSL, this is a rare case of hematochezia and anemia owing to its large size and morphology.The prevalence of SSLs is approximately 25% of all serrated polyps (SP), and ranges from 2% to 8% in average-risk patients undergoing colonoscopy. Further studies of the endoscopic morphology and biological nature of these lesions are required.In the present case, a large pedunculated polyp with a long, thick stalk was found in the rectum. Unlike the previously reported pedunculated forms of SSLs, this polyp could be misdiagnosed as a colon hamartoma or adenoma based on its morphological features and location. As previously suggested, this lesion can be a rare variant of SSLs or a precursor or result of an SSL.In conclusion, we report an unusual case of large pedunculated SSL in the rectum. The polyp was treated with endoscopic mucosal resection and pathologically confirmed as an SSL. Our findings indicate that endoscopists should always keep in mind SSLs in any location, even with unusual morphological findings.We sincerely express our gratitude to Prof Ji-Youn Sung  from the Department of Pathology, Kyung Hee University School of Medicine.SJO, JWK, and CHO designed and drafted the manuscript, and substantively revised the manuscript. All authors approved the submitted version . All authors agreed to be personally accountable for the author’s own contributions and to ensure that questions related to the accuracy or integrity of any part of the work, even those in which the author was not personally involved, were appropriately investigated and resolved, as documented in the literature.Conceptualization: Shin Ju Oh, Jung-Wook Kim, Chi Hyuk Oh.Writing – original draft: Shin Ju Oh, Jung-Wook Kim, Chi Hyuk Oh.Writing – review & editing: Shin Ju Oh, Jung-Wook Kim, Chi Hyuk Oh.


Background: Testicular torsion is a surgical emergency that needs prompt diagnosis and treatment. Point-of-Care ultrasound (POCUS) can not only establish the diagnosis but also guide the Emergency Physician in evaluating the response to manual detorsion. Case Report: We describe the case of a 13-year-old male who presented with acute scrotal pain. We demonstrate how bedside ultrasound was used to make the diagnosis of testicular torsion, guide the technique for manual detorsion, and confirm adequate return of blood flow. Our case illustrates the ease with which POCUS can be used in real time to diagnose and treat organ-threatening pathology, but more importantly, it shows how real-time POCUS was used to detorse a testicle that was refractory to the standard detorsion technique. Conclusion: The acute scrotum is a time-sensitive presentation and if testicular torsion is present, the diagnosis should be made as soon as possible. Many Emergency Departments do not have 24-hour coverage of ultrasound technicians, which would delay the diagnosis and treatment. Moreover, when manual detorsion is attempted, it often does not work because the testicle may need more than the standard 180 degree medial to lateral rotation. POCUS provides real-time analysis of return of blood flow and can thus guide further rotation, or opposite direction rotation, as needed.  Testicular torsion is a surgical emergency with a yearly incidence of 3.8 per 100,000 males under the age of 18 Pre-operative manual detorsion is the fastest way to restore blood flow to the scrotum. This maneuver involves the physical rotation of the affected testicle in the opposite direction of the torsion, most commonly medial to lateral (“open book”). Manual detorsion can improve testicular salvage, however there is wide variation in the reported success of this strategy (26-95% successful) A 13-year-old healthy male presented to the emergency department with acute onset of atraumatic right testicular pain 2 hours prior to arrival. He was afebrile and hemodynamically stable. Genital exam was remarkable for a firm high-riding right testicle, which was diffusely tender to palpation without overlying skin changes. Point-of-care ultrasound (POCUS) was performed using a high-frequency linear probe, which was placed in the transverse axis along the scrotum to obtain an image where both testicles are visible side by side . Color and pulse-wave doppler were used to evaluate testicular blood flow, demonstrating lack of flow to the right testicle, confirming the diagnosis of acute testicular torsion.Manual detorsion was then attempted, initially by twisting the right testicle medial-to-lateral on the vascular pedicle 180 degrees. Bedside ultrasound was used to reevaluate the testicle and it continued to demonstrate a lack of flow . The testicle was then rotated an additional 90 degrees, after which the patient had immediate relief of his pain and the testicle no longer felt as firm. Bedside ultrasound confirmed return of blood flow to the testicle . The urology service was consulted, and the patient was taken to the operating room for emergent orchiopexy.Testicular torsion is a surgical emergency and prompt diagnosis and treatment is necessary for testicular salvage The diagnostic accuracy of POCUS for testicular torsion when performed by emergency physicians is 95% sensitive and 94% specific We attest that our institution does not require IRB approval for case reports or de-identified clinical images and that the appropriate consent for the use of these images has been obtained from the patient/legal guardian. All authors reviewed and assisted with revisions of the final manuscript. RVN takes responsibility for the manuscript as a whole. None


Herniation of the transverse colon and volvulus of it occurring through the previous surgical drain site, presenting as intestinal obstruction, has never been reported. We present an 80-year-old female who complained of abdominal swelling for 10 years. She started developing pain abdomen for 10 days and obstipation for three days. Abdominal examination showed a tender mass in the right lumbar region, with all borders being distinct, and there was no cough impulse. There is a lower midline scar from the previous laparotomy and a small scar over the swelling (drain site). Imaging studies were diagnostic of large bowel obstruction due to the herniation and volvulus of the transverse colon through the previous surgical drain site. She underwent laparotomy, derotation of transverse colon with hernia reduction, and onlay meshplasty. She had an uneventful postoperative course and was discharged. Transverse colon herniation can occur through a previous abdominal surgical scar. The transverse colon can undergo volvulus and may present with features of intestinal obstruction. Volvulus of the herniated transverse colon through the previous surgical drain site is never known to occur. No such case has been reported in the literature so far. The typical clinical presentations of the patient with transverse colon volvulus usually present with features of intestinal obstruction. Patients who develop obstructed incisional hernia also present with abdominal pain, abdominal mass, nausea, vomiting, and obstipation ,2. ImagiAn 80-year-old female presented to the emergency department with complaints of swelling in the right flank region for the last ten years. The swelling was gradually progressive in size. The swelling increased in size on straining and reduced partially on lying down. Now she has complained of intermittent colicky abdominal pain for ten days, abdominal distension, and obstipation for three days. There was no history of vomiting, fever, jaundice, or reduced urine output. She gave a history of abdominal hysterectomy 30 years back, with re-exploration in the immediate postoperative period, probably due to gossypiboma with drain placement in the right flank region.At the presentation, she was dehydrated; her pulse rate was 110 beats per minute, blood pressure was 110/80 mm Hg. Abdominal examination revealed swelling of 20 cm x 20 cm over the right lumbar region, tender, and firm in consistency. All borders of the swelling were distinct and round. There was no cough impulse in the swelling. There is a lower midline scar from the previous laparotomy and a small scar over the swelling (drain site). Bowel sounds were exaggerated. Per rectal examination revealed collapsed rectum.3, and normal platelet count. The liver function test (LFT) and renal function test (RFT) were within normal limits. Arterial blood gases were within normal limits. X-ray abdomen showed a few dilated bowel loops on the right side of the abdomen with air-fluid levels. There was no air under the diaphragm .


Mucinous cystadenocarcinoma (MCAC) with malignant ascites is rare. We report a case of a 28-year-old woman who presented with epigastric pain. The ascites in the Douglas fossa was identified at a nearby gynecology clinic. Computed tomography showed a multiloculated cystic lesion (9.5 × 6.4 cm) in the tail of the pancreas, which was diagnosed as mucinous cystic neoplasm on imaging. Staging laparoscopy was performed, and rapid cytology of ascites revealed adenocarcinoma, leading to a diagnosis of unresectable MCAC. Subsequently, combination chemotherapy with gemcitabine plus S-1 was initiated. Although there were no remarkable changes in the imaging findings, the peritoneal dissemination node was not consistently recognized in any of the imaging findings, and distal pancreatectomy was performed. A peritoneal dissemination node was not observed in the laparotomy findings, but the peritoneal lavage cytology was positive. The postoperative pathological result was non-invasive MCAC, and the ascites was suspected to be caused by cyst rupture. The patient has been recurrence-free, including the reappearance of ascites, for > 8 years after adjuvant therapy with S-1. Although careful follow-up will be required in the future, the very good prognosis in this case suggests that MCAC with malignant ascites without obvious peritoneal dissemination should be considered for surgical resection. Pancreatic mucinous cystic neoplasm (MCN) is a rare cystic neoplasm that is typically located in the pancreatic body or tail of middle-aged women . In 2000When MCAC is localized in the pancreas, surgical resection is recommended and mostly associated with a favorable prognosis . PalliatWe report the case of a patient with MCAC with malignant ascites who underwent surgical resection after combination chemotherapy with gemcitabine plus S-1 (GS). Although surgical resection is not indicated for pancreatic ductal carcinoma with malignant ascites, in this case, surgical resection was performed because of the absence of peritoneal dissemination and the young age. As a result, the patient has had no recurrence, including no reappearance of ascites for more than 8 years after postoperative adjuvant therapy.A 28-year-old woman had occasionally presented epigastric pain. Approximately 10 months after the onset of the epigastric pain, the patient visited a nearby gynecology clinic because of abnormal vaginal bleeding. Transvaginal ultrasonography at the clinic revealed ascites in the Douglas fossa, and she was referred to the gynecology department of a nearby university hospital. Douglas fossa puncture for the cytology of ascites was performed, and adenocarcinoma was suspected (Papanicolaou class IIIb). The gynecological examination revealed no abnormal findings in the uterus and ovary; however, the computed tomography (CT) scan revealed ascites in the pelvis and a well-defined 9.5 × 6.4 cm multilocular cystic lesion in the tail of the pancreas  on days 1 and 8 plus 100 mg/day of S-1  from days 1 to 14, repeated every 3 weeks per course, were initiated. Due to grade 4 neutropenia during the first course, the GEM dose was reduced to 80% from the second course. At the end of the sixth course (4.5 months later), the best overall response to chemotherapy on the Response Evaluation Criteria in Solid Tumors version 1.1 was stable disease on contrast-enhanced CT. Furthermore, although ascites was still observed, a slight decrease in serum CA19-9 was observed (from 30.7 to 9.9 U/mL). Positron emission tomography–CT (PET–CT) that was performed to re-evaluate the feasibility of surgery, demonstrated 18F-fluorodeoxyglucose uptake only in the solid component of the pancreatic tail cyst, and the maximum standard uptake value (SUVmax) was 2.94 , were 1.0 ng/mL and 30.7 U/mL, respectively. The cancer board with the department of surgery was carried out, and the diagnosis reached at our institution was also unresectable MCAC. Subsequently, combination chemotherapy with 1,000 mg/mDistal pancreatectomy and lymph node dissection were performed for curative resection in July 2013. The cyst adhered to the spleen and stomach, and the spleen and part of the serosa were resected together. The peritoneal dissemination node was not revealed in the laparotomy findings, and cleaning of the peritoneal cavity was performed using saline. Peritoneal lavage cytology revealed adenocarcinoma. The specimen was a multilocular cystic tumor measuring 6.0 × 9.0 × 5.5 cm that was filled with pale yellow mucus Fig. . The mulAfter surgery, she was treated with 120 mg/day of S-1 from days 1 to 28, repeated every 6 weeks per course, for four courses, as adjuvant therapy. She has been free of recurrence, including the no reappearance of ascites for more than 8 years after adjuvant therapy with S-1.MCAC has been reported to have a better prognosis than typical pancreatic ductal adenocarcinoma . HoweverStandard chemotherapy for MCAC has not been established; thus, it has been treated as pancreatic ductal adenocarcinoma. Chemotherapy with GEM for unresectable MCAC has been successful in some reports, but not in others , 8. ThisIf malignant ascites is present, surgical resection is not indicated for pancreatic ductal adenocarcinoma. Consequently, at the beginning, the cancer board of our institution judged this case as not an indication for surgical resection, due to the presence of malignant ascites. However, the risk–benefit balance of the treatment option continued to be examined during chemotherapy with GS, and finally, surgical resection was performed. There have been no previous reports of surgery performed in patients with MCAC, who have been preoperatively proven to have malignant ascites. However, the very good prognosis in this case suggests that surgical resection should be considered in the absence of peritoneal disseminated nodes on PET–CT and other imaging or staging laparoscopy findings, with careful follow-up after surgery. In fact, in this case, it seems to be also possible to carry out the upfront surgical resection without initiating chemotherapy, because the disseminated node has not been identified from the beginning. When the existence of peritoneal dissemination cannot be ruled out and there is a concern about an upfront surgical resection, it is important to administer chemotherapy, such as GS, as preoperative adjuvant therapy and to reassess the need for surgical resection based on imaging evaluation after a certain period.Surgical resection should be considered even in patients with MCAC with malignant ascites, as long as peritoneal disseminated node is not identified. If peritoneal dissemination cannot be ruled out and there is a concern about upfront surgical resection, continued evaluation of the feasibility of surgery during chemotherapy, such as GS, may be considered as a treatment strategy for MCAC with malignant ascites.


Subpial haemorrhage is a rare cause of seizures in term neonates. A 3-day-old male infant, born at term with no history of perinatal hypoxia, presented with seizures and unremarkable physical examination in the interictal state. Imaging demonstrated left temporal subpial haemorrhage with the classic ‘yin-yang sign’ on MRI. The patient was subsequently diagnosed with factor XIII deficiency. Follow-up at 6 months and 12 months revealed encephalomalacia in the previous haemorrhagic areas with normal developmental milestones. The risk factors and pathophysiology of this entity are unique compared with other forms of extra-axial haemorrhage.Few cases of subpial haemorrhage have been described in the literature due to their relative rarity, which is estimated at approximately 4% according to pathological reports.This case report describes subpial haemorrhage in the left temporal region and the typical ‘yin-yang sign’ described in the recent literature in a term neonate presenting with seizures. There was also intraparenchymal and intraventricular extension at presentation. Encephalomalacia of the previous haemorrhagic areas was evident at follow-up imaging six months later, but no breakthrough seizures or motor developmental delay was reported on follow-up after one year.A 3-day-old term male infant presented with seizures and lip-smacking. There was no fever nor any signs of recent trauma. The perinatal history was unremarkable, and the baby was delivered via spontaneous vaginal delivery. His older brother was 12 years old with no relevant medical history. There were two prior abortions – one spontaneously at the third month of gestation and another that was induced with mifepristone after the 45th day of amenorrhea. No identifiable cause was found for the prior spontaneous abortions.Routine blood investigations revealed no significant abnormality with normal prothrombin time (PT), activated partial thromboplastin time (aPTT) and international normalized ratio (INR), normal sepsis screen and negative cerebrospinal fluid (CSF) culture. Imaging was requested to determine the cause of the seizures.An initial CT scan indicated a large hyperdense localised extra-axial haematoma in the left temporal region with intraparenchymal and intraventricular extension and mild hydrocephalus. The underlying cortex was hypodense, likely reflecting ischaemic changes . The MRIThe patient was diagnosed with subpial haemorrhage, and therapy with oral phenobarbitone was commenced. Electroencephalogram (EEG) revealed spikes suggestive of generalised seizure activity. The baby improved symptomatically and was discharged after a week of observation with the recommendation for detailed clotting factor profile assays to exclude coagulopathies.Results of the clotting profile revealed factor XIII deficiency. On follow-up examination at six months of age, microcephaly was observed with the head circumference two standard deviations below normal. However, there was no significant motor neurological deficit for this age nor any breakthrough seizures. MR imaging revealed extra-axial cystic changes in the left temporal region with no new haemorrhage . The undAt the age of one year, the child had started walking with support and was able to articulate words expected for his age. There were no seizures nor any evidence of motor delay. The child continues follow-up and remains seizure free.8 The pial sheath in the perivascular spaces around the intracerebral arteries is in direct continuity with them in their subarachnoid course as well.9 A strong trabecular meshwork is present along the vessels in the subarachnoid space but is absent in the subpial space, which may result in increased fragility of the subpial vessels.4Extra-axial haemorrhages are typically classified according to their location as subdural, subarachnoid or extra-dural. Subpial haemorrhage is a rare entity and can be radiologically confused with subarachnoid haemorrhage. The pia mater is the innermost, highly vascular layer of the meninges covering the entire brain and the spinal cord. It invests both the brain and spinal cord until the conus medullaris, following which it tapers and forms the bulk of the filum terminale.10 The first cases of subpial haemorrhage were described in neuropathology specimens by Friede in 1975 wherein he described them as dissecting underneath the pia mater without dissecting into the subarachnoid space or the neuroparenchyma. He thought it to be a variant manifestation of respiratory distress syndrome and assumed that it began with an injury to the superficial glial cells in the molecular layer of the cortex.7 The same has also been described independently as ‘superficial cortical tears’ by Lindenberg in 1969, six years before Friede’s seminal work.11 These subcortical clefts or tears are now attributed to being a part of the secondary cascade due to impaired venous drainage and not necessarily related to traumatic events.12Subpial haemorrhage has been uncommonly documented and is usually ascribed to trauma or as part of child abuse – the ‘shaken baby syndrome’.13 Likely, this anatomy, combined with the absence of the trabecular mesh in the pial space, produces a potential pathomechanism by which venous injury can decompress superficial haemorrhage into the subpial space without haemorrhage in the subarachnoid space.14 A case report in 2015 also described subpial haematoma in the Sylvian fissure in an adult and attributed it to bleeding from small vessels in the subpial space.15 This may also provide the mechanism for the intraparenchymal extension that was observed in the presented case.The mechanisms by which haemorrhage occurs in the subpial space have not been definitively discovered but current theories regarding venous thrombosis have the most validity. Okudera et al. detailed the venous anatomy of the cerebral hemisphere using roentgenograms of pathological brain specimens in 1999 and described the subcortical and superficial medullary veins that run superficially and penetrate the cortex to join the pial vessels.16 Their well-localised nature and ‘yin-yang sign’ (representing the hypointense haemorrhage and hyperintense underlying cortex on T2 and diffusion-weighted MR sequences) are characteristic differentiators from other extra-axial counterparts. Additional major differentiating features are the presence of localised mass effect from the haemorrhage and ischaemic changes of the underlying cortex, which are not seen with subarachnoid or subdural haemorrhages.17Subpial haemorrhages typically appear as a well-circumscribed extra-axial haemorrhage, usually seen adjacent to a gyrus. They may be bilateral, depending on the underlying pathology (such as coagulopathy), and are usually located adjacent to the cerebellum or temporal regions.4 A cohort study of 17 patients by Cain et al. noted medullary venous congestion or thrombosis adjacent to the areas of subpial haemorrhage and suggested a venous pattern of ischaemia as the cause rather than birth trauma.6Roth et al. described non-traumatic subpial haemorrhage in a population of 10 patients with ages ranging from 53 years to 80 years, and observed that most of their patients recovered completely, suggesting an excellent prognosis.18 However, this was not evident in the current case study.Dabrowski et al. conducted the largest retrospective cohort study, to date, including 31 neonates with subpial haemorrhage. The majority were term babies (55%) with the subpial haemorrhages being commonly multifocal and if isolated, located in the temporal lobe. About 77% of cases had other haemorrhages, the most common being intraparenchymal haemorrhage. The majority of cases had no intervention but were followed up clinically and with imaging; most had some form of developmental delay on follow-up after at least 1 year.Subpial haemorrhage is a relatively uncommon finding in clinical practice. Knowledge of its appearance, together with the recently described ‘yin-yang sign’ on MRI, will prevent misdiagnosis and exclude other extra-axial haemorrhages that might have different prognoses. The diagnosis of subpial haemorrhage should also initiate a search for background coagulopathies, which may be the underlying cause as in this case.


Congenital perineal hamartomas are rare, and reports of prenatal ultrasound diagnosis are limited. Perineal hamartomas are usually associated with other structural malformations, which complicate the therapeutic regime.We report a case of perineal hamartomas associated with rectal duplication in a female fetus. A review of the literature on similar cases was also presented. A fetus was first diagnosed with a perineal mass at 33 weeks of gestation using ultrasound examination in our hospital. Two-dimensional ultrasonography showed a hyperechoic mass resembling a scrotum in the perineum of the fetus. The pedicle connected the mass to the fetal anus. The masses were excised after birth, and perineal hamartomas were confirmed by pathological diagnosis. Rectal duplication, an associated malformation, was found during the surgery. The rectal duplication cyst was removed at the same time.Congenital perineal masses are rare and are usually associated with urogenital and anorectal malformations. Prenatal ultrasound should be used to assess the position and relationship between the mass and perineal organs, and to exclude other combined deformities. According to available reports, congenital perineal masses, including lipomas, hamartomas, and hemangiomas, are rare . AlthougA 34-year-old woman, gravida 2, para 1, was transferred to Shengjing hospital of China Medical University at 33 weeks of gestation. At 24 weeks of pregnancy, the patient was informed that she was pregnant with a boy during an ultrasound examination at the district hospital. However, during the 32-week ultrasound examination, the doctor found abnormalities in the perineum of the fetus. A detailed ultrasound examination was performed at 33 weeks in Shengjing hospital, and a perineal scan revealed that the fetus was female, not male. Two-dimensional ultrasound showed a hyperechoic mass (1.7 × 1.2 cm) resembling a scrotum in the perineum of the fetus . The peden bloc excision of the cystic mass and the posterior wall of the rectum was performed. Rectal duplication was confirmed by histopathological examination. At the time of writing this report, the patient was 2.5 years old, and recovered well after surgery without other abnormalities.A female neonate was delivered, weighing 3,240 g at GA 40 + 2 weeks. Her Apgar scores were 10 and 10 at 1 and 5 min, respectively. Perineal examination revealed two soft spherical masses protruding from the patient's anus and connected by a pedicle. The masses were 2.5 × 1.5 cm and 1.4 × 0.7 cm in size . The neoPerineal masses include lipomas, hamartomas, hemangiomas, and other types of tumors . Perineain utero on comprehensive sonographic scanning and no other abnormalities on MRI after birth. However, rectal duplication was observed during postnatal surgery. This suggests that prenatal ultrasound examination was limited to the examination of the perineal organs and structures. Pediatric surgeons should be aware of the possibility of other associated malformations, even if prenatal examinations are normal.Only a few studies have reported congenital perineal masses. We found 23 similar cases (including our case) are summarized in Here, we report a case of perineal hamartomas associated with rectal duplication, for the first time. Rectal duplication is uncommon. Patients with rectal duplication present with chronic constipation, acute urine retention, and other clinical manifestations . In our The embryogenesis of congenital perineal masses can be poorly interpreted. However, congenital perineal masses can destroy the continuity of the sacrococcygeal region, leading to combined defects of the anorectal and genitourinary organ systems , 19. In Three-dimensional ultrasound plays an important role in auxiliary diagnosis. We used the three-dimensional surface imaging technology to vividly display the perineal masses, which also clearly showed the origin and location of the mass and its relationship with other organs. Three-dimensional ultrasound is also conducive to enhancing diagnostic confidence and facilitating better communication with patients.In conclusion, prenatal ultrasound examination plays a leading role in distinguishing the origin of perineal hamartomas and evaluating the associated abnormalities. This could provide an efficient basis for clinical counseling and postnatal treatment.The original contributions presented in the study are included in the article/supplementary material, further inquiries can be directed to the corresponding authors.Written informed consent was obtained from the individual(s), and minor(s)' legal guardian/next of kin, for the publication of any potentially identifiable images or data included in this article.YZ and LC were responsible for the original draft preparation and editing. MZ, WM, and YZ participated in data collection. LC, DW, and ZY were responsible for conceptualization, manuscript editing, and manuscript review. All authors have read and approved the final manuscript.


Foot and ankle tumors are relatively rare. Nevertheless, the calcaneus is a prevalent location accommodating various lesions. Reactional periostitis of the lateral wall is rarely encountered but can potentially mimic a wide variety of tumors. We present a case of excessive proliferation due to chronic compression of the peroneal tendons against the calcaneus in a female patient with a history of diminished foot control, treated successfully by tumor excision and peroneal restoration via the tubularization technique. This study aimed to underline the mimicking potential of reactional periostitis and its effect on the peroneal tendons and hindfoot motion. Calcaneal tumors and tumorous entities are relatively rare, comprising approximately 3% of foot and ankle osseous lesions . TherefoReactional periostitis, an inflammatory entity of the periosteum provoking woven bone deposition, may affect the calcaneal peroneal tubercle (CPT). This may be a result of various conditions such as trauma, arthritic changes, infection, tumors, and certain drugs (e.g. teriparatide). The appearance of a periosteal reaction is determined by the intensity, aggressiveness, and duration of the underlying insult . A wide We present a case of excessive CPT osseous proliferation due to chronic compression of the peroneal tendons against the calcaneus in a female patient with a history of diminished foot control following spinal surgery. The purpose of this article is to underline the mimicking potential of reactional periostitis and its effect on the peroneal tendons and hindfoot motion.A 65-year-old female patient was admitted due to a painful mass on the lateral aspect of her right hindfoot. Regarding her medical history, she reported a right drop foot following a spinal surgery 10 years ago, which eventually improved over time. Seven years ago, she noticed a lump on the lateral side of her foot, around the calcaneus, that was asymptomatic. Nevertheless, the same mass was reported enlarging through time, causing gait difficulties and becoming painful during ambulation.Clinical examination indicated reduced eversion strength (3+ out of 5), but no further signs suggesting a drop foot lesion. Meanwhile, the pain was elicited upon direct palpation of the peroneal tendons. Plain radiographs and CT images showed a bony overgrowth originating from the CPT, separating the peroneal tendons and pushing them off the calcaneus Figure .A subsequent MRI revealed diffuse bone marrow edema accompanied by a low-intensity lesion on T2-weighted images , identifying the peroneal tendons and dislocating them off the peroneal tubercle. An osteotome was used to remove the proliferative tubercle at the same level of the calcaneal lateral wall. Both peroneal tendons were debrided of scar tissue and sutured using the tubularization technique with absorbable sutures . The excPostoperatively, a weight-bearing cast was applied for three weeks. There were no complications regarding wound healing. Sutures were removed three weeks later and the patient was allowed to fully weight-bear without restrictions after the fourth week. Histological evaluation of the lesion revealed chronic inflammatory synovial proliferation and bone remodeling, typical findings of reactional periostitis Figure .At the six-month follow-up visit, eversion strength had significantly increased, with the patient reporting substantial pain improvement, even after prolonged walking. More specifically, the preoperative American Orthopaedic Foot And Ankle Society (AOFAS) score of 71 points (out of 100 maximum) significantly improved to 95 points, while a preoperative visual analog scale (VAS) pain score of 8 points (out of 10 maximum) improved to 2 points. Eversion strength was estimated to be 4+ from 3+ (out of 5) before her operation. In addition, a follow-up MRI  -9. OsteoOn the other hand, osteochondromas (cartilage-capped bony projections or outgrowth on bones' surface) share similar radiological features with the peroneal tubercle overgrowth, including a continuous cortex with a medullary cavity that flows into the lesion. In addition, there may be a stalk (pedunculated) or a broad connection to the cortex (sessile) along with cartilaginous coverage. In our case, the lesion was located on the lateral calcaneal wall with diffuse bone marrow edema. Nevertheless, neither a demarcated cavity nor chondral overlay was identified on the MRI.We did not obtain a preoperative biopsy on this patient. The reason is that, as concluded by the above differential diagnosis, both her medical history and the radiological features were indicative of a benign tumor. Therefore, although excluding the diagnosis of an osteochondroma was not definite preoperatively, the treatment remains the same as in reactive periostitis ,13. In aThe peroneal tubercle separates the peroneus longus and brevis tendons. Hypertrophy may occur in response to trauma, physical overactivity, or inflammatory changes related to peroneal tendon activity, resulting in peroneal misalignment. In patients with pes cavus, this chronic pressure of peroneal tendons against the lateral calcaneal wall may result in reactive periostitis and thus hypertrophy of the peroneal tubercle -18. In oIn conclusion, despite being a rare entity, it should be noted that the distinction of foot and ankle tumors is crucial as they demonstrate a vast mimicking potential. Suspicion of a possible malignant lesion should always be raised, while knowledge of the local anatomy and the clinical and radiological features of each specific tumor is essential to apply the appropriate treatment without delay. Reactional periostitis, usually produced by chronic peroneal pressure against the lateral calcaneal wall, although a benign condition, can be a severely limiting factor in patients' everyday routine, causing constant pain and gait abnormalities. It is vital to be adequately diagnosed and treated early, as removing the tumor and restoring the damaged peroneal tendons can improve life quality by inducing pain relief and improving the patient's lever arm.


Teaching Point: Benign granulomatous prostatitis mimics prostate cancer on MRI. Peculiar urological history of a patient undergoing prostate MRI helps sorting out that differential, such as a treatment with topic Bacilli Calmette-Guerin instillations. The patient is a 66-year-old man. He was treated 22 months ago for bladder urothelial neoplasm (pT1G3) by resection and topic bacilli Calmette-Guerin (BCG) instillations (Oncotice). At follow-up consultation, the digital rectal exam (DRE) revealed firmer prostatic left lobe and trans-rectal ultrasound showed a suspect hypoechogenic lesion at the left prostatic base. Prostatic-specific-antigen (PSA) level was slightly increased (4.77 ng/ml). A multi-parametric magnetic resonance imaging (mp-MRI) of the prostate revealed two focal lesions in the left peripheral zone. Both presented nodular high signal intensity on b1500 diffusion-weighted imaging , low appGranulomatous prostatitis is a well-known but uncommon condition representing only 3.3% of all benign inflammatory lesions of the prostate. The incidence in the general population varies between 0.5 and 1.86% (1). Mean age of presentation is 61 years. GP may be idiopathic (nonspecific) or secondary to multiple causes including some specific urinary tract infective agents , iatrogenic , malacoplakia associated with recurrent infection or other systemic granulomatous diseases like sarcoidosis. Idiopathic GP remains the most frequent type, seen in 69 to 77.7% of all cases [At medical inspection, DRE can reveal a stiffer area. PSA may be elevated, and the patient can show hematuria. All these findings may also be present in prostate cancer (PCa). Moreover, some studies reported the coexistence of GP and carcinoma in 10–14% of cases [On mp-MRI, GP appearances may completely overlap those of prostatic adenocarcinoma, showing nodular shape, low signal on T2-weighted imaging, high signal intensity on diffusion weighted imaging and low ADC. There is no defined ADC value which can differentiate both pathologies. Perfusion study may also be confounding. Nevertheless, some patients with GP present with a rim-enhancement related to abscess or caseous formation.Clinical information is the key to evoking this diagnosis in case of secondary GP. Treatment should be adapted to the specific cause. In idiopathic GP, conservative and symptomatic treatment is administrated followed by surgery if ineffective.


Isolated sublingual hematoma is a rare complication seen in trauma, severe uncontrolled hypertension, dental operations, bleeding diathesis, and the use of dual antiplatelet and anticoagulant agents. In advanced and neglected cases, a sublingual hematoma may interfere with the patient's airway, causing suffocation and fatal airway obstruction. Our objective was to present a case of iatrogenic isolated sublingual hematoma in a 70-year-old business man, heavy smoker with a history of hypertension. Furthermore, to report the literature review, and to organize treatment strategies to reduce the rate of progression of the hematoma. In addition to recommend or advice a strategic plan to prevent this complication during carotid stenting.This case report has been reported in line with the SCARE Criteria. We represented a case report of an iatrogenic isolated mild/moderate sublingual hematoma in a 70-year-old business man. This hematoma developed as one of the rare complications of endovascular internal carotid artery revascularization because of injury to the sublingual branch of the lingual artery during wire manipulations or its advancement. This hematoma was treated by conservative treatment without any intervention.Immediately after internal carotid artery stenting procedure, the patient developed a sudden onset of painful swelling in the floor of the mouth. The hematoma showing ecchymotic submucosal swelling underneath the tongue in the floor of the mouth. If it became enlarged, it may push the tongue against the palate and blocking the airway, causing serious airway obstruction. Fortunately, the swelling is isolated (has no extension to any side) and limited to the root and middle third of the tongue but, not extending to its tip. Its development most probably due to injured atherosclerotic sublingual branch of the lingual artery during the procedure.The first step in management should be prompt airway management. Conservative treatment took place without any further intervention. To date, there is no consensus about the management regarding the hematoma itself. Mostly, clinicians start with observation for spontaneous resolution. When conservative treatment is not appropriate, surgical intervention must be performed. However, electively secure the airway is the main objective for treatment. •We presented a case report of iatrogenic sublingual hematoma as a complication of internal carotid artery stenting.•The first step in management should be prompt airway management.•As the patient was stable without any respiratory and airway compromise, conservative treatment took place without any further intervention, by closed observation and watchful waiting.•When conservative treatment is not appropriate or failed, surgical intervention must be performed.•To date, there is no consensus about the management of the hematoma itself.•Finally, and according to our best of knowledge, this is the first and a unique case that reported an isolated sublingual hematoma following internal carotid artery stenting. Sublingual hematoma is considered as a rare and potentially fatal complication that may develop after carotid artery stenting (CAS). The tongue is a highly vascular structure with extensive anastomotic network. Therefore, there is a potential risk of hemorrhage and consequently hematoma formation following local trauma or iatrogenic injury during any related procedures 2This work has been reported in line with the SCARE Criteria 3Bleeding into the sublingual space may elevate the tongue and increase the dead space in the floor of the mouth resulting in a non-infectious life-threatening upper airway obstruction. This picture was first reported in 1976, and known as pseudo-Ludwig's phenomenon Although, there are no literature reports regarding the application of either ice or a cold packs or adrenalin packs. They may be considered accepted methods of causing local vasoconstriction of the bleeding vessel/s but, not reducing the size of the hematoma. They may be used under the chin and applied for 10 to 20 min at a time. The periprocedural risk of stroke or death following carotid artery stenting (CAS) was estimated as 4.7% (range 2–9%) from a Cochrane review of more than 5000 CAS procedures 4We reported this case to endorse the importance of postoperative care of carotid artery stenting procedures and to introduce such rare yet a potentially fatal complication. The first line management of isolated sublingual hematoma, should be prompt airway patency, and it must be considered as the main goal for treatment. To date, there are no reported guidelines regarding the management of such complication. However, most clinicians start their management conservatively, in the hope of spontaneous resolution. When conservative treatment is not appropriate, surgical intervention must be performed. Finally, this is the first reported isolated sublingual hematoma after carotid artery stenting according to the best of our knowledge. On failure of conservative treatment and watchful waiting, surgical intervention in the form of tracheotomy must be adopted to electively secure the airway. In the future, and to avoid this complication, we recommend gentle manipulations and exchange of the guide wires during carotid artery stenting. However, empirical administration of unfractionated heparin during the procedure must be avoided, and provided according to the body weight or it may be given at a suboptimal dose.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.Not commissioned, externally peer-reviewed.The authors declare that, they obtained permission from the ethics committee in our institutions.None declared.Ahmed Mousa.None.Ahmed Mousa: Data curation, Formal analysis, Investigation, Methodology, Validation, Writing, original draft, Writing, review and editing.Bassam A. Khalil: Data curation, Formal analysis, Investigation, Methodology, Validation, Writing, original draft, Writing, review and editing.None declared.


Intussusception of the cecum due to acute appendicitis is rare condition.A 17-year-old male patient presented to our hospital with a chief complaint of right lower abdominal pain, which had lasted for two days. Computed tomography (CT) revealed a “target sign” from the cecum to the ascending colon, leading to a diagnosis of cecocolic intussusception. Colonoscopy revealed an erythematous, edematous, and internally distorted cecum in the ascending colon, which was difficult to repair with air insufflation. Laparoscopic surgery was performed to remove the bowel obstruction. Repositioning of the invaginated cecum was difficult due to the presence of a hard and edematous colic wall. Therefore, laparoscopic ileocecal resection was performed to release the obstruction. The pathological diagnosis was appendicitis and abscess within the cecum wall, with no malignant findings.In our case, intussusception was considered to have caused thickening of the intestinal wall of the cecum due to inflammation of the appendix, and the thickened area became the leading point.Considering that malignancy is a frequent leading point in adult patients with intussusception, a preoperative endoscopic examination is important for minimizing bowel resection. •Most cases of intussusception outside of childhood are caused by tumors, but inflammation of the appendix or cecum can be a cause.•If malignancy is not the cause of intussusception, bowel resection can be minimized.•In order to determine the appropriate extent of resection, an accurate preoperative diagnosis is necessary, keeping in mind the possibility that appendicitis or inflammatory changes in the cecum may be the leading point. This work has been reported in line with the SCARE criteria 23/μl, C-reactive protein: 1.6 mg/dl). Computed tomography (CT) showed a swollen appendix and appendicolith in the middle of the appendix  were children (<18 years of age), one patient (12.5 %) was in his 20s, and one patient (12.5 %) was in his 70s. The majority of cases involved young patients. Therefore, in childhood, it may be related to the fixation and mobility of the cecum and ascending colon to the retroperitoneum. In addition, in adults, it may be due to a lack or loosening of fusion of the cecum to the retroperitoneum.Among the eight reported cases of cecocolic intussusception caused by appendicitis, the most common symptom was abdominal pain associated with intestinal obstruction (87.5 %), followed by nausea and vomiting (12.5 %), and fever (25.0 %). Bloody and gelatinous stools were also observed in pediatric patients. The most common symptoms of intussusception are reported to be due to bowel obstruction: cramping abdominal pain (71 %), nausea and vomiting (68 %), abdominal distention (45 %), and tenderness (60 %); however, these symptoms are often absent in adults Abdominal ultrasonography and CT are commonly used for the diagnosis of intussusception. Its characteristic findings include the “target sign” and “doughnut sign” in transverse sections, and the “hay fork sign” and “pseudo kidney sign” in longitudinal sections In our case, the pathological findings revealed submucosal edema and inflammatory cell infiltration in the appendix, which led to a diagnosis of acute appendicitis. In previous reported cases appendicitis was also diagnosed pathologically . There wIn eight cases of cecocolic intussusception caused by appendicitis, laparoscopic surgery was performed in 7 cases (87.5 %); only one pediatric case (12.5 %) was relieved by conservative treatment. Among the 7 patients who were treated by surgery. Laparoscopic appendectomy and bowel resection were performed in 4 and 3 patients, respectively. In cases in which intussusception and bowel obstruction are difficult to remove, it is necessary to consider bowel resection.4We experienced a case of cecocolic intussusception caused by acute appendicitis. Surgical treatment was safely performed based on an accurate and appropriate preoperative diagnosis.None.Not applicable in this case report as it is not a research study.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.Not commissioned, externally peer-reviewed.All the authors contributed to diagnose and treat the patient. Naoya Kimura and Masatsugu Hiraki contributed in drafting the manuscript. Toshiya Tanaka and Kenji Kitahara supervised and made the final approval of the manuscript. All authors read and approved the final manuscript.None.Toshiya Tanaka, Department of Surgery, Saga Medical Center Koseikan.The authors declare no conflicts of interest in association with the present study.


Multiple Endocrine Neoplasia 1 (MEN1) syndrome is a genetic condition arising from a mutation of the MEN1 gene resulting in neuroendocrine tumor formation. Patients with MEN1 are at a higher risk of developing Zollinger-Ellison syndrome (ZES) due to the growth of neuroendocrine tumors called gastrinomas that release gastrin leading to hypersecretion of acid in the stomach resulting in severe ulcerative disease of the upper GI tract. Our case is a 42-year-old female with newly diagnosed MEN1 syndrome, presenting with acute abdominal pain and dyspepsia refractory to medical management including proton pump inhibitors (PPI) and H2 antagonists. ZES was biochemically confirmed with a secretin stimulation test and dotatate positron emission tomography/computed tomography (PET/CT) revealed multiple areas of hyper-metabolic activity within the gastrinoma triangle. However, no discrete masses could be appreciated on endoscopic ultrasound (EUS) or CT imaging that could provide a target for surgical intervention. This case elucidates not only the difficulty of gastrinoma localization in medically refractory ZES but also reinforces the need to screen patients with MEN1 presenting with acute abdominal pain and dyspepsia for ZES. MEN1 syndrome is a rare autosomal dominant condition defined by the development of neuroendocrine tumors throughout the body. The most common sites involved include the parathyroid, pituitary, and pancreas . About 2A 42-year-old female with a seven-month history of persistent hypercalcemia ranging between 11 to 13 mg/dl presents to the emergency department with right flank pain. CT of the abdomen and pelvis showed nephrolithiasis as well as masses in the pancreatic tail and right adrenal gland Figure , 1B. On There are two main classifications of ZES, sporadic and MEN1 associated . DiagnosFirst-line management for ZES is acid suppression therapy with PPI or H2 blockers with gastric pH goals of greater than four to prevent erosion and ulceration of the GI tract mucosa . In patiWith the emergence of acid suppression medical therapy with PPI and H2 blockers, there has been a radical reduction in the need for acid-reducing surgical interventions in patients with ZES associated with MEN1 . NeverthGastrinomas can be very difficult to localize and EUS is generally recommended when surgical excision is being considered, as it is superior to CT imaging modalities in tumor localization . This caThe liver is the most common location of metastatic spread for gastrinomas, followed by the axial skeleton. Gastrinomas have high malignant potential and approximately half of all patients with ZES will have liver metastases upon initial diagnosis . MetastaMEN1 is an autosomal dominant disorder that is due to mutations in the MEN1 tumor suppressor gene resulting in neuroendocrine tumor growth throughout the body. There is a strong correlation between ZES occurring in the setting of MEN1. There are no general guidelines on the treatment and management of medically refractory ZES in the setting of MEN1 in which gastrinoma cannot be localized as surgical excision is not possible until a discrete mass is identified. Given this association, as well as the high morbidity and mortality of ZES, physicians should have a low threshold to screen for ZES in MEN1 patients complaining of gastric reflux or dyspepsia with a gastrin level, chromogranin A level, and EGD in order to allow for early diagnosis and reduce the potential of developing life-threatening complications of ZES.


Paragangliomas of the gallbladder are exceptionally rare. To date, only a few cases of this disease have been reported globally, and the majority were found incidentally during surgery. Although complete resection can achieve a curative effect, specific targeted drugs may have survival benefits for patients with potential recurrence and metastasis risks.quaque die (every day)]}, and no recurrence was observed during the regular follow-up.A 48-year-old woman was scheduled for anatomical central hepatectomy due to the discovery of a liver mass. Surprisingly, a gallbladder tumor accompanied by intrahepatic invasion was found rather than primary liver lesions during the operation. Postoperatively, the lesion was confirmed to be a paraganglioma originating from the gallbladder with intrahepatic invasion detectable on histopathology. After surgery, the patient was treated with a new targeted drug, surufatinib {200 mg, q.d. [Gallbladder paraganglioma is rare and occult, and surgeons do not know it well, so it is easily misdiagnosed before surgery. Postoperative pathological examination is the gold standard for diagnosis.Given that the tumor contained abundant blood sinuses, the early and continuous enhancement of dynamic enhanced CT scanning was its characteristic manifestation. We presented a case in which a primary gallbladder paraganglioma was identified accidentally in a patient who was misdiagnosed with a liver lesion before surgery. Based on our experience in this work, the en bloc resection technique in combination with surufatinib might have a survival benefit to patients at risk of potential recurrence or metastasis; however, further follow-up observations are needed. In recent years, compared with other solid tumors, the incidence rate of neuroendocrine neoplasms (NENs) has increased significantly. In general terms, NENs can be divided into the following classifications: well-differentiated neuroendocrine tumor (NET), poorly differentiated neuroendocrine carcinoma (NEC), pheochromocytoma (PHEO), and paraganglioma (PARA) . ParaganIn terms of the symptoms and histological features of “pheochromogenic reaction”, sympathetic paraganglioma is closely related to pheochromocytoma. Given the similarities between sympathetic paraganglioma and pheochromocytoma, the treatment options are essentially the same . Paragan18F-FDG (fluorodeoxyglucose F 18) PET-CT] scanning, which revealed no other hypermetabolic lesions. Collectively, the final diagnosis was primary gallbladder paraganglioma with partial liver invasion. In view of the abundant blood supply of the tumor and its potential for malignancy, we recommended that the patient take surufatinib, a new oral tyrosine kinase inhibitor developed completely independently in China at 1 month after surgery. Surufatinib has dual anti-angiogenic and immunomodulatory activity, and has been shown to be effective in clinical trials in non-pancreatic neuroendocrine tumor patients. We regularly followed up with the patient for half a year; her general condition was fine. Serum NSE levels showed a continuous downward trend  showed a poorly defined low-density lesion in the middle lobe of the liver (mainly located in the S4b and S5 segments), which was significantly enhanced, and was suspected to be an angiogenic liver tumor Figure 1Paragangliomas are neoplasms originating from the paraganglia, which develop from neural crest cells and form the scattered nerve endocrine system around or within the autonomic nervous system, which extends from the skull base to the pelvic floor. In general, paragangliomas in the adrenal medulla are called pheochromocytoma, while those outside the adrenal gland are designated extra-adrenal paragangliomas, also known as neurogenic NENs . Extra-aAt present, we still know very little about primary gallbladder paragangliomas. Since the first report of a gallbladder paraganglioma by Miller et al. in 1972 Table 1 A thorough and detailed pathological examination after the operation was critically important for the diagnosis and differential diagnosis of NENs. The difference between NETs/NECs  and PHEO/PARA  is that the former has keratin expression, which was negative for spectral keratin in our patient . MicroscCurrently, it is commonly believed that about 35%–40% of paragangliomas/pheochromocytomas (PPGLs) are genetically related . TherefoIn light of the fact that extra-adrenal tumors are more commonly clinically aggressive than adrenal tumors, and that in this case the tumor partially invaded the liver, although we had implemented a surgical scheme similar to the radical resection of a gallbladder carcinoma, postoperative recurrence problem is always a concern. As a new type of oral tyrosine kinase inhibitor, independently developed in China, surufatinib  has the In conclusion, primary paragangliomas of the gallbladder are extremely rare in clinical settings and little is known about them. High levels of vigilance towards preoperative imaging is required for the detection of lesions that show significant and persistent enhancement early on the enhanced CT within the gallbladder. The characteristic immunohistochemical expression profile and the detection of pathogenic genes are critically important for diagnosis. In the era of targeted therapy, surufatinib might be a more effective treatment option for such tumors that have a risk of metastasis and recurrence, although more high-grade, high-quality clinical studies are needed to prove this in the future. We hope that through this case, more doctors can deepen their understanding of this rare disease and learn from our experiences and lessons.The raw data supporting the conclusions of this article will be made available by the authors, without undue reservation.The studies involving human participants were reviewed and approved by Ethics Review Board of the Inner Mongolia People’s 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.YX, SW, and LX collected imaging data and wrote the article, JD performed histopathological analysis of tissue sections, and LZ and LX reviewed and revised the manuscript. All authors contributed to the article and approved the submitted version.


A 73‐year‐old man taking lanthanum carbonate for hemodialysis showed progressing gastric mucosal changes with lanthanum deposition. Regular examination revealed concurrent gastric carcinoma. The extent and depth of its invasion were ambiguous because of the surrounding lanthanum deposition. Furthermore, there could be other potent carcinomas, and curative laparoscopic gastrectomy was performed. It is difficult to determine the clinical depth and extent of involvement by the gastric carcinoma in cases of lanthanum deposits with hemodialysis. Such cases require more careful preoperative evaluation and therapeutic strategy. Moreover, sometimes those features might resemble those of carcinoma. In such cases of gastric cancer with lanthanum deposition, careful pretherapeutic diagnosis is warranted.Pathologically, lanthanum depositions usually appear as subepithelial collections of histiocytes or small foreign body granulomas, accompanied by intestinal metaplasia, regenerative changes, and foveolar hyperplasia.There are also few reports regarding gastric cancer concurrent with lanthanum depositionUnlike previous cases in which surgical resection was performed, in this case, it was difficult to clearly determine the depth and extent of invasion by gastric adenocarcinoma; however, the surrounding ambiguous mucosal changes with lanthanum deposition were clearly observed. Further, such cases require careful diagnosis and a definitive therapeutic strategy. Herein, we present a case of laparoscopically resected gastric adenocarcinoma with concurrent lanthanum deposition in which the depth and extent of invasion by the tumor was difficult to determine for precise preoperative diagnosis.2A 73‐year‐old man had been on peritoneal dialysis for 9 years for chronic renal failure due to immunoglobulin A nephropathy and had switched to hemodialysis 3 years before the surgeries.He had been taking lanthanum carbonate for 7 years. Several months after lanthanum carbonate initiation, regular endoscopy revealed white granular lesions on the mucosa of the gastric body and fundus Figure . The res3We report a rare case of laparoscopically resected gastric adenocarcinoma with concurrent lanthanum deposition. In this case, we chose surgical resection because of the clinical depth of the tumor which was classified as T1b; possibility of occurrence of other potential carcinomas with lanthanum deposition; and ambiguous boundary between the malignant tissue and mucosal changes with lanthanum deposition.There are a few reports of surgically resected gastric cancer with lanthanum deposition, as summarized in Table In gastric adenocarcinoma concurrent with lanthanum deposition, the findings of lanthanum deposition by endoscopy are occasionally similar to carcinoma, and it might be difficult to diagnose the precise depth and extent of the tumor and mark the boundary between the neoplasm and lesions. The findings of lanthanum deposition have been usually described as shiny, bright white, and of varied sizes and shapes, from small and flat to elevated plaques.The relationship between malignancy and lanthanum deposition in gastric cancer concurrent with lanthanum deposition is unknown. Yabuki et al.Herein, we presented a rare case of laparoscopically resected gastric adenocarcinoma concurrent with lanthanum deposition because of the difficulties in diagnosing the precise clinical depth and extent of the tumor, in determining the possibility of occurrence of other neoplastic lesions with lanthanum deposition, and in defining the ambiguous boundary between the carcinomatous and noncarcinomatous changes in the lesion. The clinical significance and relationship between neoplasm and lanthanum deposition have been suggested, but enough evidence has not been reported. Our results add to this growing body of data and will aid in clarifying this relationship.KH performed the surgery and observed the patients, selected the associated data, and edited the manuscript accordingly. SS also performed the surgery and took care of the patient. YO analyzed the specimens and supervised the pathology. KM performed the surgery on the patient as a supervisor. KI and KK performed the treatment of the patient as gastroenterologists. TH, NS, TN, and HM supervised and revised the article. All the authors have read and approved the final manuscript.This research received no specific grants from any funding agency in the public, commercial, or not‐for‐profit sectors.All the authors declare no competing interests.This study was performed in accordance with the ethical standards of the Japan Surgical Society.Written informed consent was obtained from the patient for publication of this case report and accompanying dates and images.


Spontaneous retroperitoneal bleeding is a rare occurrence that might have catastrophic implications. We present a 58-year-old male with a 4-day history of progressively worsening left-sided flank pain due to retroperitoneal hemorrhage from a nonfunctional renal paraganglioma. Abdominal contrast CT scan was helpful in locating the tumor, estimating tumor size and extent of bleeding, visualizing the interaction between the tumor and the surroundings, and ruling out any potential metastasis; however, it lacked specificity in identifying the origin of the mass, needing histologic investigation for a conclusive diagnosis. MRI was not available at our center. We report a rare case of spontaneous retroperitoneal bleeding as a complication of an unusual nonfunctional renal paraganglioma, which was initially misdiagnosed as renal cell carcinoma but later confirmed by postoperative histopathology. Extra-adrenal autonomic paragangliomas are rare neuroendocrine tumors that develop from the paraganglia. These are tiny organs made up primarily of neuroendocrine cells generated from the embryonic neural crest that can release catecholamines . ParaganSpontaneous retroperitoneal hemorrhage is a well-known but uncommon condition characterized by retroperitoneal bleeding without a prior history of trauma. It may strike fast and quietly, with catastrophic consequences if not diagnosed and treated on time. Clinically, it presents with acute abdominal pain, mainly in the affected flank, palpable mass, and hypovolemic shock in severe blood loss . SpontanA 58-year-old man presented to the emergency department complaining of left-side abdominal pain for four days. The pain began suddenly and moved to the left groin and testes. However, it was only associated with constipation through passing flatus. There was no history of vomiting, sweating, palpitation, or hematuria. There was no past medical history of hypertension or diabetes.On examination, the patient was pale (anemic), had no pedal oedema, and had normal blood pressure of 120/70 mm/Hg. There were distended abdomen and tenderness at the left hypochondrium, left lumbar fossa, and suprapubic region.H. pylori, negative; stool occult blood test, negative; and urine analysis, no detection of erythrocytes.Hematology parameters were as follows: HB, 6.0 g/dl 13.0–18.0); leukocytes, 16.47 (4.0–12.0); platelets, 170 × 10(9) 150–450); ESR, 38 mm/hr [3.0–18.0;0; ESR, 3On radiological examination, ultrasound showed a left renal complex lesion that is predominantly solid with some cystic components seen at the left upper renal pole. It demonstrated mild internal vascularity on the Doppler study.Subsequent computed tomography (CT) of abdomen and pelvis showed an exophytic upper pole cortical mass (red arrowhead) in the left kidney (red arrow) measuring 7 × 4.5 × 7.3 cm in transverse, anterior-posterior, and craniocaudal diameter, respectively . On contThe patient was then scheduled for surgery, which included a left nephrectomy with complete mass excision, and a total of 3.5 liters of blood and clots were removed from the peritoneum . The hilHistology: on low magnification, the left side shows normal-looking renal tubules and the right side shows the fatty tissue with round blue cells consistent with tumor cells . On highParagangliomas/pheochromocytoma are rare neuroendocrine tumors arising from chromaffin cell tumors located at any extra-adrenal site along the sympathetic or parasympathetic nervous system and account for 15 to 20% of chromaffin cell tumors . The incRetroperitoneal extra-adrenal paragangliomas make up the majority of extra-adrenal paragangliomas, accounting for 85% of all extra-adrenal paragangliomas . The renDiagnosis of paraganglioma necessitates the combination of clinical symptoms, biochemical evaluation of catecholamine production by the tumor, and tumor location , 24. TheThe biochemical diagnosis of hyperfunctional paraganglioma or adrenal pheochromocytoma is based on excessive excretion of catecholamines and their metabolites. The 24 h urinary excretion of total metanephrine and catecholamine metabolites was determined to have a sensitivity of 74% and 89.9%, respectively . The EndAs per our case, preoperative screening for catecholamines was regrettably not performed because the patient initially presented with acute abdomen and severe anemia instead of catecholamine-related symptoms and was also normotensive. In biochemically silent abdominal paragangliomas , the lack of catecholamine secretion is not related to catecholamine storage or secretion mechanisms, but rather to a deficiency in catecholamine synthesis that results in a near-complete lack of releasable reserves. The problem is caused by a lack of tyrosine hydroxylase , 31. AndIncidental discovery of nonfunctional paragangliomas has increased over the last two decades using high-resolution abdominal imaging techniques prior to the onset of symptoms. 29.4% of the 201 total study population was diagnosed incidentally to have pheochromocytoma/paraganglioma . ImagingThe classical “salt-and-pepper” appearance of paragangliomas on T1- and T2-weighted images is due to the presence of punctate areas of low signal intensity corresponding to flow voids from tumor vascularity, and hyperintense signal corresponding to areas of hemorrhage in the tumor. This sign has been described more in head and neck paragangliomas than in trunk paragangliomas, and it is also neither sensitive nor specific, as it can be seen in any hypervascularized tumor, specifically angiofibromas .Spontaneous rupture of nonfunctioning retroperitoneal paraganglioma is rare and quite difficult to be diagnosed initially as it presents with severe abdominal pain that mimics other acute surgical conditions , 40. OurSpontaneous hemorrhage induced by paraganglioma can present as an abdominal catastrophe, which tends to be lethal. In a review of literature, so far 50–53 cases have been identified with ruptured pheochromocytoma worldwide and mortality rate of 32–34% globally , 42. AltHistologically, paraganglioma is usually composed of solid nests known as “zellballen” of round-to-oval or elongated cells with abundant granular amphophilic or basophilic cytoplasm, within a vascular stroma, but they may also have acidophilic cytoplasm . MalignaA concept of risk stratification is used to describe the metastatic potential of pheochromocytoma and paraganglioma . Malignaα-receptor blocker prior to surgery [Complete surgical excision is the treatment of choice for extra-adrenal retroperitoneal paragangliomas, and recurrent or metastatic disease but should be carried out on the basis of correct drug preparation of  surgery . In the  surgery ; this ca surgery . There wFurthermore, risk stratification is required to tailor the follow-up protocol after complete resection of paraganglioma. The overall rate of recurrent disease was found to be 0.98 events/100 person-years, and primary tumors larger than 5 cm have a higher risk of recurrence .Retroperitoneal pheochromocytoma/paragangliomas are rare tumors that are usually benign with good prognosis. The clinical picture of the patient might range from asymptomatic to a dramatic life-threatening occurrence such as sudden acute bleeding from the tumor. Incidental discovery of the pheochromocytoma and paragangliomas on imaging is becoming an increasingly essential mode of their diagnosis. Radiological differential diagnoses are often inadequate or challenging in such cases, especially for tumors originating from kidneys. Therefore, it is important for clinicians and radiologists to be more aware of pheochromocytoma/paragangliomas and their various appearances in order to avoid potential surgical complications without further evaluation prior.


Neuroblastic neoplasms (NN) include ganglioneuromas (GN), ganglioneuroblastomas (GNB), and neuroblastomas (NB). They generally arise in childhood from primitive sympathetic ganglion cells. Their incidence in adults, especially among elderly, is extremely low.This is the case of a 74-year-old woman with history of abdominal pain, weakness and night sweating since several months. Blood pressure was normal. CT-scan showed a 10 cm left adrenal mass, without other pathologic findings. An open left-sided adrenalectomy was performed. Recovery was uneventful with hospital length of stay of 8 days. Based on morphological, immunohistochemical, and molecular features the diagnosis was a nodular GNB. A positron emission tomography (PET) performed 6 weeks after the resection did not show any residual tumor or distant metastases. The patient was followed-up with annual clinical and radiological exams.This case presentation, associated with a review of the literature, illustrates the importance to include NN in the preoperative differential diagnosis of adrenal tumors in adults and highlights the need for multidisciplinary patient work-up and management. Neuroblastic neoplasms (NN) are a heterogeneous group of tumors deriving from the sympatho-adrenal lineage of the neural crest forming the peripheral sympathetic nervous system. They include a wide spectrum of neoplasms with various degrees of cellular maturation and biological aggressiveness –4. GanglNB are the most frequent extra cranial solid tumors in early childhood, (8–10% of childhood cancers), while GNB are very rare, including in the pediatric population  –7. In adBecause of the rarity of NN in adults, the natural history in this population is unknown and there are no established treatment guidelines. To the best of our knowledge, no case of adrenal GNB in elderly patients has been reported in literature until now . In the A 74-year-old woman consulted her GP with history of abdominal pain, weakness and night sweating for months. She was on prednisone and pregabalin for fibromyalgia. The patient had no history of hypertension. The physical examination showed a palpable mass in the left upper quadrant, blood pressure was normal. A thoraco-abdominal contrast-enhanced CT-scan, showed a 10 × 10 × 8 cm mass at the upper pole of the left kidney , 2 and nSerum catecholamines showed elevated nor-metanephrine  and methoxytyramine , but normal metanephrine levels  . Urine cAn open left adrenalectomy was performed through a left subcostal laparotomy. The definitive diagnosis was nodular GNB .Recovery was uneventful with hospital stay of 8 days. Following our multidisciplinary endocrinology board, the staging was completed with PET-CT, which showed no distant lesions. It was programmed a clinical follow up at 1 month after the surgery with a favorable clinical course and a resolution of all symptoms. The patient was followed with annual clinical exams and CT-scans.The clinical presentation of NN is very heterogeneous and depends on the site of origin and the presence of metastatic disease . ApproxiNB and GNB can secrete catecholamines, and their derivates such as vanillylmandelic acid and homovanillic acid. Determination of catecholamine levels and their serum metabolites is important for the initial screening, diagnosis, differential diagnosis , and for the evaluation of treatment efficacy . Neurobl123-MIBG-scan or FDG PET-CT, are essential in the assessment of NB and GNB to define the extent of disease at diagnosis and to follow the treatment response  and the International Neuroblastoma Risk Group Staging System (INRGSS), are summarized in According to the INRGSS and INSS, our patient qualifies as a stage L1 and a stage 1, respectively. Adults with L1 disease experience an OS of 94, 90, and 69% at 3, 5, and 10 years, respectively . The mosNN patients with a high quantity of neurotrophin receptors, found on the surface of normal nerve cells and some neuroblastoma cells, especially the nerve growth factor receptor TrkA, may have a better prognosis .In older children, but not in infants and adults, MYCN amplification represents a negative prognostic factor . The ampOutcomes of NN in adults are generally poor, because half of the patients have a stage IV NB at the time of diagnosis , 13. 45%In children, treatment of NB and GNB is well-defined but remains demanding. Surgical resection is the mainstay for low-risk pediatric cases, whereas high-dose chemotherapy with stem cell rescue, radiation therapy and immunotherapy are indicated in high-risk patients . In chilRegarding treatment, the presented patient had surgery without chemotherapy or radiation therapy. In adults, due to rarity, there are no standardized guidelines or treatment protocols for NN . AccordiFollow-up with urinary catecholamine levels and imaging is necessary for all pediatric patients treated for NB and GNB . As funcThere are no clear guidelines concerning imaging-based follow-up, but the frequency of visits and duration of observation period depend on age, stage and localization of the tumor. In some children, ultrasound may be used for surveillance, while CT, MRI or even MIBG-scan can be necessary for adults or advanced-stage metastatic disease , 36. FDGGNB should be included in the preoperative differential diagnosis of adrenal tumors in adults. In the absence of specific guidelines, management of adrenal NN in elderly is surgical following pediatric protocols and retrospective observational studies.The original contributions presented in the study are included in the article/supplementary material, further inquiries can be directed to the corresponding author/s.Written informed consent was obtained from the individual(s) for the publication of any potentially identifiable images or data included in this article.PD wrote the manuscript and the abstract. RD was the first corrector. MM and TZ were the second correctors. TZ was the coordinator. SL and CG added the pathology section and the pictures. MB-P added some informations concerning her medical specialties. All authors read and approved the final manuscript.Open access funding was provided by the University of Lausanne.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.All claims expressed in this article are solely those of the authors and do not necessarily represent those of their affiliated organizations, or those of the publisher, the editors and the reviewers. Any product that may be evaluated in this article, or claim that may be made by its manufacturer, is not guaranteed or endorsed by the publisher.