Intraperitoneal pseudocysts are rare and may be difficult to differentiate from other malignant neoplasms. Reports of occurrences are mainly associated with long-term intraperitoneal catheter use, intraperitoneal catheter infections, or major pelvic surgery in females, although there are few reported incidences without prior trauma. We present a case of a male patient found to have a 19 × 15 × 9 cm intraperitoneal pseudocyst with other multiloculated areas and a history of a right inguinal hernia repair. After a thorough review of the literature, this is the first multiloculated nonpancreatic pseudocyst reported in a male patient. Intraperitoneal pseudocysts are rarely reported and are mainly secondary to inflammatory insults such as ventriculoperitoneal shunts, catheter infections, or even major pelvic surgery in females –8. Only A 58-year-old male presented to his primary care physician with central abdominal pain and distension. His past medical history was significant for hypertension, hyperlipidemia, and end-stage renal disease secondary to hypertensive kidney disease requiring hemodialysis. This was performed via arteriovenous fistula starting 8 years prior to being seen. He denied any history of peritoneal dialysis and his only abdominal surgery was a right inguinal hernia repair approximately 4 years before. Unfortunately, the operative report was unavailable to us and he was unsure if mesh was used. CT of the abdomen and pelvis with oral and intravenous contrast showed a 17.2 × 14.6 × 17.3 cm large pelvic cystic tumor with separate loculated areas in the right upper abdomen adjacent to the gastric antrum. A smaller fluid collection in the right midabdomen between the mesocolon of the proximal transverse colon/hepatic flexure and small intestine, ascites, and thickened omentum were also seen Figures . The priHe underwent a bronchoscopy that showed extrinsic compression from the loculated effusion. Washings showed atypical cells but no malignancy was identified. His information was reviewed and discussed in our gastroenterology multidisciplinary conference and a decision was made to proceed with surgical exploration and resection of the large intra-abdominal tumor. Given the possibility for mucinous tumor, surgical debulking as well as potential intraperitoneal chemotherapy was also discussed.During surgery, the tumor was found to be adherent to the omentum and anterior abdominal wall and was unable to be separated requiring resection of a portion of peritoneum. This was suspected to be close to the region of the hernia repair, although it was difficult to discern if scar tissue or tumor caused the inability to separate the tumor from the peritoneum. No mesh was encountered in this space. The mass was also adherent to the right colon but was able to be separated with careful dissection. It was not attached to the appendix, but this was removed given the original concern for mucinous ascites. The tumor appeared encapsulated with a tan-yellow capsule and multiple thick adhesions were encountered . MultiplFinal pathology revealed an intact large cystic mass measuring 19 × 15 × 9 cm . GrosslyThe patient recovered well postoperatively without complication and was discharged home on postoperative day 6. Most recent imaging performed approximately 3 months later showed no intraperitoneal recurrence, but he required a right thoracentesis for a recurrent pleural effusion. CEA was not rechecked given that no carcinoma was found.We present a case of a giant multiloculated intraperitoneal pseudocyst in a male patient. Few prior reported cases without a history of surgical intervention have been found in the literature –11. Repode Perrot and colleagues proposed a classification of mesenteric cysts that included (1) cysts of lymphatic origin; (2) cysts of mesothelial origin; (3) cysts of enteric origin; (4) cysts of urogenital origin; (5) mature cystic teratoma; and (6) pseudocysts . Older cThe largest series of intraperitoneal cysts included 16 patients with mesenteric cysts reported over 14 years . This seThe clinical significance of these tumors should not be underestimated. Their large size can result in a wide range of symptoms and they may be difficult to differentiate from a neoplastic process, especially when multiloculated areas are found. Our patient only complained of distension and abdominal pain; however, they can cause a wide variety of symptoms including constipation, bowel obstruction, or even an acute abdomen secondary to infection, bleeding, rupture, volvulus, or even bowel ischemia , 17. Add


Pineoblastoma is a rare malignant tumor of the central nervous system (CNS), which arises from the parenchyma of the pineal gland. It is characterized by aggressive clinical behavior and frequent metastases along the craniospinal axis. Extraneural metastases may occur due to surgical seeding of tumor cells beyond the dura and/or hematogenous spread, ventriculoperitoneal shunting, or through Batson’s plexus. To our knowledge, only six documented cases of intraosseous metastases of pineoblastoma are described in the literature.A 23-year-old female patient presented with clinical and radiological symptoms of a pineal tumor causing secondary hydrocephalus. After initial surgical treatment, chemotherapy, and local radiotherapy with craniospinal irradiation, she developed multiple metastases affecting the anterior skull base, intracranial meninges, frontal bone, and finally, the entire vertebral column. The patient received surgical treatment for the anterior skull base metastasis, repeated irradiation of the neuraxis, radiosurgical and radiotherapeutic procedures, and chemotherapy. The patient survived 57 months after the primary disease manifestation and died of multiple metastases.This presented case is the first known description of metastasis of pineoblastoma in the anterior cranial base. Multiple intracranial metastases were suppressed using CyberKnife radiation treatment and chemotherapy until massive involvement of spinal column occurred. Interestingly, no signs of brain radiation necrosis after repeated radiation treatments were observed, and the patient developed only moderate neurocognitive decline. Pineoblastoma is a rare malignant tumor of the central nervous system (CNS), which arises from parenchyma of the pineal gland and primarily affects the pediatric population. Pineal tumors comprise 0.5-1% of all intracranial tumors, and 15-32% of pineal tumors originate from pineal parenchyma. Pineoblastoma represents 25-50% of pineal parenchymal tumors ..3].In our observation, intraosseous metastases  occurred after surgical resection, local radiotherapy combined with whole brain and spinal cord irradiation, and six cycles of chemotherapy.We agree with Constantine, et al.  that wheThe demonstrated case is the seventh published intraosseous metastasis of pineoblastoma and the first known description of metastasis in the anterior cranial base. Multiple intracranial metastases were suppressed using CyberKnife radiation treatment and chemotherapy until a massive involvement of the spinal column occurred. Our patient survived 57 months after primary disease manifestation and died of mushrooming metastases.Our observation demonstrates the possibility of local control of macroscopic disease using conventional and stereotactic radiotherapy, surgery, and chemotherapy. Two courses of whole brain radiation and prolonged chemotherapy led to only moderate neurocognitive decline. Interestingly, we did not observe any signs of brain radiation necrosis after repeated radiation treatments of numerous leptomeningeal metastases. Another issue that deserves special attention is the development of multiple intraosseous metastases  at the late stage of the disease. Unfortunately, the exact cause of lethal outcome is unknown, but we may suppose that the patient died from massive tumor dissemination.


Caliber-persistent artery (CPLA) of the lip is a common vascular anomaly in which a main arterial branch extends to the surface of the mucous tissue with no reduction in its diameter. It usually manifests as pulsatile papule, is easily misdiagnosed, and is observed more frequently among older people, suggesting that its development may involve a degenerative process associated with aging; CPLA is also characterized by the loss of tone of the adjacent supporting connective tissue. Although the diagnosis is clinical, high-resolution Doppler ultrasound is a useful noninvasive tool for evaluating the lesion. This report describes the case of a 58-year-old male patient who complained of a lesion of the lower lip with bleeding and recurrent ulceration. The patient was successfully treated in our hospital after a diagnosis of CPLA and is currently undergoing a clinical outpatient follow-up with no complaints. A caliber-persistent artery (CPLA) of the lip is a primary arterial branch that enters the submucosal tissue and shows no reduction in its diameter , 2. ThisThis type of abnormal artery was first described in the oral cavity in 1973 by Howell and Freeman, who designated it as “prominent inferior labial artery” , and thiThe lesion presents as a papular, arcuate, or linear elevation with a normal or bluish pale color. In general, the artery becomes inconspicuous when the lip is stretched. The characteristic feature of this disorder is lateral and vertical pulsation. The lesion is initially asymptomatic and usually diagnosed during a routine clinical examination. The patient may occasionally notice an increase in the pulse volume in the lip .Microscopically, the lesion is characterized by a normal muscular artery with an irregular lumen and a size that is unusual for its location in the submucosa , 12.CPLAs occur almost exclusively on the lip mucosa and can affect both the upper and lower lips , 13, 14,Some cases may be associated with ulceration of the mucosal lining, and other rare cases may occur adjacent to a lip squamous cell carcinoma. CPLAs are occasionally associated with ulceration , 8, 9 anThe factors that contribute to lip ulceration or erosion in CPLAs include accidental or chronic vascular atrophy or ectasia in the elderly, ischemia due to the increased pressure of a pulsating superficial submucosal artery, medications, chronic actinic cheilitis, and arteriosclerosis .Although there have been reports of CPLAs in the stomach and other areas of the gastrointestinal tract, with some cases involving hemorrhage and death, no cases of bleeding in a CPLA of the lip have been reported. However, severe blood loss is possible due to trauma to the lip or excision without the identification of this tortuous artery. Therefore, recognition of the presence of this condition in the oral cavity is extremely important due to the risk of bleeding during oral and maxillofacial surgery .Cases of CPLAs found in the lower and upper lips have involved dilatation of the inferior labial artery and superior labial artery, respectively , 13, 15.A 58-year-old male patient presented with swelling in the lower lip with bleeding and recurrent ulceration. The patient complained of swelling for two months before the diagnosis when he noticed the presence of a traumatic ulcer in the region. The swelling increased in size after it was first noticed and was accompanied by pain on palpation. The patient exhibited no systemic changes.A clinical examination indicated the presence of a bullous lesion along the left lower lip vermilion without well-defined margins and with the following characteristics: soft consistency, smooth surface, having same color as the mucosa, being approximately 1 cm at its largest diameter, and presence of a fistula Figures . The lesThe differential diagnoses included mucocele, traumatic fibroma, canalicular adenoma, leiomyoma, and hemangioma.The patient reported no past trauma or damaging oral habits; in addition, no clinical evidence suggestive of the presence of vascular proliferation in the adjacent tissue was found. Considering these data, the patient was clinically diagnosed with a CPLA of the lip.The pulsating area was surgically explored under local anesthesia, and an approximately 1 mm CPA was detected in the submucosal layer. Subsequently, the artery was isolated using 2.0 cotton thread, and its most superficial portion was excised. Upon cleaning and irrigating the surgical cavity with 0.9% saline, the mucosa was sutured with 4.0 silk thread Figures –8. AThe surgical specimen was sent for anatomopathological examination and identified as an arterial section, which, considering its unusually superficial position, provided a conclusive diagnosis of a CPLA in the lip mucosa .The descriptive term CPLA was initially used to describe a vascular anomaly in the stomach submucosa. The first studies on the subject indicated that the vascular anomaly in the mouth was associated with mucosal ulceration, possibly due to the ischemia caused by the vascular pulsation in the mucosa , 8, and In 1985, Marshall and Leppard described a case of CPLA that also affected the lower lip and was associated with an ulcer with a progression period of five months. These authors performed surgery with safety margins because the ulcer was clinically compatible with squamous cell carcinoma; however, a histopathological examination indicated the presence of a benign lesion.In that report, the clinical aspects included a pulsatile tubular lesion on palpation, and an ulcer was observed in the mucosa upon visual inspection, in contrast to the results of other studies that found no association with ulcers , 10, 12.Although an angiographic examination was not performed, the case reported herein showed characteristics similar to those described by Howell, Freeman, and Jaspers , 9: unilUlceration is thought to occur as a result of “pulsating pressure exerted by the artery on the epithelium” in the labial mucosa. Senile atrophy and/or solar damage have been listed as possible contributing factors. Solar damage is evident but not relevant to our case because the labial mucosa is usually exposed to this factor. The patient had no history of trauma but was a smoker. However, considering that the lesion was located in the occlusal plane, it was chronically exposed to occlusal trauma with recurrent bleeding. The patient became aware of his injury when the ulcers began to appear. A congenital CPLA malformation of the lip was likely but tended to remain unnoticed until mucosal ulceration began to occur. In previous reports, the youngest patient with a lip ulcer diagnosed as CPLA was 56 years old, and the average age was 73 years. Howell and Freeman [Clinically, the differential diagnosis of CPLA includes varicose veins and hemangioma when the lesion is visible, in addition to the presence of nonvascular entities, including mucocele and fibroma , 13. A cDifferent theories have been postulated to explain the etiology of CPLA. Some authors believe that trauma, cigarette smoking, or the pressure of the cigarette against the lip may contribute to the decrease in the thickness and composition of the connective tissue that supports the arteries, leading to arterial dilation , 13. ThiThe treatment of choice is surgical removal of the surface vessel, primarily in patients at risk of mucosal biting, those prone to falls from their own height , and those uncomfortable with the pulsation. Lovas et al.  reportedLovas et al.  reviewed


Twenty years old young was referred to our department due to painful inguinal mass. The mass was diagnosed as torsion of third testis which was treated by orchiectomy. Polyorchidism is a rare entity with increased risk for malignancy and torsion. Polyorchidism is a rare condition; it happens due to an embryological abnormal division of the genital ridge .These supernumerary testes are at increased risk to develop malignancy and torsion , 2.Triorchidism is the most common form, but also 4/5 testes have been reported in literature.The supernumerary testes are usually intrascrotal .The minority of all reported cases are inguinal and some of them even retroperitoneal.The third testis is usually more mobile and more prone to torsion .Ultrasound colour plays a crucial role in diagnosing and following up this entity. MRI plays a role when the diagnosis is in doubt or when suspecting an associated malignancy.Some patients after bilateral orchiectomy remained reproductive because of missing the undiagnosed third undecsended testis. In fact this happens because some of the supernumerary testis has attachment to a draining epididymis and vas deferens –3.Twenty years old young, was referred to our ultrasound unit because of a history of two-day painful right inguinal mass. The patient which is usually healthy had no fever in these two days.Physical examination revealed a tender mass in the right inguinal area, the patient had no fever, and laboratory tests were in normal range.Ultrasound of the inguinal area showed an inguinal oval 1.6 cm, echogenic mass with no flow in it that resembles small testis . UltrasoEmbryologically polyorchidism can be classified into four types . In typeTriorchidism is the most common type of polyorchidism and presents with two testes on one side  and one testis on the other side. Rare case of polyorchidism with three homolateral testes on the right side and absent testis on the left side has been reported , 4.Splenogonadal fusion is a rare congenital anomaly that may sonographically resemble polyorchidism . In thisPolyorchidism is a rare genitourinary abnormality, and torsion is one of its associated complications; however, the diagnosis of polyorchidism with or without torsion can be made readily with ultrasonography when one is aware of this entity .Sonographic features of torsion are homogenous hypoechogenicity of testis with absent flow on color Doppler study. MRI can play a role in diagnosing torsion; signs in MRI are increased signal intensity on T1 and decreased signal intensity on T2 weighted sequences .Not every inguinal mass is a hernia or a lymph node.Polyorchidism is a rare congenital anomaly.Torsion is a complication of this entity.Ultrasound Doppler plays a crucial rule in diagnosing.If you do not think about it, you will not mention it in your diagnosis.


Vast majority of bowel obstruction is due to postoperative adhesions, malignancy, intestinal inflammatory disease, and hernias; however, knowledge of other uncommon causes is critical to establish a prompt treatment and decrease mortality. Xanthomatosis is produced by accumulation of cholesterol-rich foamy macrophages. Intestinal xanthomatosis is an uncommon nonneoplastic lesion that may cause small bowel obstruction and several cases have been reported in the English literature as obstruction in the jejunum. We report a case of small intestinal xanthomatosis occurring in a 51-year-old female who presented with one day of copious vomiting and intermittent abdominal pain. Radiologic images revealed jejunal loop thickening and inflammatory changes suggestive of foreign body obstruction, diagnostic laparoscopy found two strictures at the jejunum, and a pathologic examination confirmed a segmental small bowel xanthomatosis. This case illustrates that obstruction even without predisposing factors such as hyperlipidemia or lymphoproliferative disorders. Small bowel obstruction corresponds to approximately 20% of patients who present with acute abdomen and are admitted for surgery . Among t3/μL (5–10 × 103/μL) with neutrophilia 84% (30–40%); CT scan of the abdomen , macroglobulinemia, and muciphages . In our Intestinal xanthomatosis is a rare entity but it should be considered as one of the etiologies of clinically significant obstruction and must be included even if the patient has no history of hyperlipidemia or lymphoproliferative disorder. In order to implement the best treatment options if diagnosed, clinicians must extend workup to rule out lipid storage diseases, dyslipidemia, lymphoproliferative disorders, and infections .


Inflammatory pseudotumor (IPT) of the liver is a rare benign lesion. A case of IPT of the liver found in association with a malignant gastrointestinal stromal tumor (GIST) is reported.18Fluorine-fluorodeoxyglucose positron emission tomography (FDG-PET) showed strong uptake in the liver. A diagnosis of liver metastasis was made and partial hepatectomy was performed. Microscopic examination showed that the tumor was an IPT.A 74-year-old man was admitted to our hospital for a liver tumor. He previously underwent rectal amputation for a malignant GIST. Computed tomography (CT) revealed a low-density area in the liver and dynamic contrast-enhanced MRI (EOB-MRI) showed that the tumor was completely washed out in the delayed phase. Differential diagnosis between IPT and malignant neoplasms is difficult. Moreover, FDG-PET revealed strong uptake in the tumor. To our knowledge, this is the first patient reported to have an IPT in association with a rectal GIST. This patient is discussed along with a review of the literature. Inflammatory pseudotumor (IPT) is a rare benign tumor that may develop in various organs. However18fluorine-fluorodeoxyglucose positron emission tomography (FDG-PET) showed strong uptake in the liver, so a diagnosis was made of metastatic liver tumor of malignant GIST. IPT of the liver in association with GIST is very rare, with only two cases, including the present case, having been reported. Herein, we present our patient along with a discussion of the literature.The medical history of the present case revealed that he had previously undergone a sacroabdominal rectal amputation for malignant GIST of the rectum. Moreover, A 74-year-old man was admitted to our hospital for examination and surgical treatment of a liver tumor. His medical history revealed that he had undergone a sacroabdominal rectal amputation for malignant GIST of the rectum at age 65 years. The resected specimen showed a tumor with intraluminal growth measuring 2 cm × 2.5 cm, with a center spot ulcer. The UICC TMN classification of GIST was T2N0M0. Upon histological examination, morphologic analysis suggested a spindle-cell GIST, and the diagnosis was confirmed by immunohistochemical (IHC) investigation. MIB-1 was strongly positive, but the surgical margin was negative. The final diagnosis was high-grade rectal GIST (mitotic index > 5/50 high power field). After surgery, the patient did not want to receive adjuvant chemotherapy and he underwent regular follow-up in our hospital according to National Comprehensive Cancer Network (NCCN) guidelines. The patient did not receive any other adjuvant therapy. The patient had never been outside of Japan.Nine years after surgery, follow-up CT revealed a 14-mm low-density area in segment 8 of the liver. The periphery of this area was nonhomogeneously enhanced by contrast medium and appeared isodense in the late phase compared to surrounding normal liver tissue Figure . There wIPT of the liver is an uncommon, benign, tumor-like lesion that sometimes mimics a malignant tumor, particularly metastatic cancer or cholangiocarcinoma. IPT of et al. reported the frequencies of GISTs as follows: stomach (60%), jejunum and ileum (30%), duodenum (4 to 5%), rectum (4%), and colon and appendix (1 to 2%)[Rectal GIST is relatively rare compared to other types of GIST. Miettinen 1 to 2%). GIST is1 to 2%). In the 1 to 2%). Based oTreatment for hepatic IPT remains controversial. Spontaneous regression occurs in some cases,14, and et al. have hypothesized that the correlation may be related to inflammation[et al. suggested that biopsy was not sufficient to diagnose IPT of the liver, because the histological appearances of inflammatory pseudotumors and GISTs are particularly difficult to differentiate[et al. summarized the experience of 45 IPT cases[et al. insisted that histological diagnosis was necessary to make an accurate diagnosis of IPT. Percutaneous biopsy is useful for diagnosis, but possible coexistence of malignancy is sometimes not ruled out[et al. concluded that hepatectomy is preferable because it not only minimizes the risk of a biopsy-related complication , but it also abolishes the possibility of IPT recurrence[To our knowledge, this is the first patient reported to have an IPT associated with a rectal GIST. One previous report described a patient with IPT associated with a small bowel GIST. In bothammation. The litammation. In addirentiate. In contrentiate. In the PT cases. They inuled out. Furtheruled out-25. So Ncurrence. HoweverIn conclusion, we experienced an extremely rare case of IPT of the liver associated with rectal GIST. In general, it is difficult to distinguish IPTs from malignant tumors by radiologic studies. Moreover, in the present case, FDG-PET showed strong uptake in the lesion. Since only two reports, including the present case, have described IPT in conjunction with GIST, further analysis of the etiology based on the accumulation of additional cases is required in the future. Surgeons should be aware of the possible association between these conditions, as illustrated in the present case.Written informed consent was obtained from the patient for the publication of this case presentation and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.18Fluorine-fluorodeoxyglucose positron emission tomography; IHC: Immunohistochemical; SMA: smooth muscle actin; NCCN: National Comprehensive Cancer Network; CRP: C-reactive protein; CEA: carcinoembryonic antigen; CA19-9: carbohydrate antigen 19-9; AFP: α-fetoproteinIPT: Inflammatory pseudotumor; GIST: gastrointestinal stromal tumor; CT: computed tomography; MRI: magnetic resonance imaging; EOB-MRI: gadolinium ethoxybenzyl diethylenetriamine pentaacetic acid; FDG-PET: Yoichi Matsuo has no conflicts of interest to declare.YM was responsible for the writing. MS, TS, and MM participated in data collection. KT, TS, and TH participated in literature searching. TS, HT and HT carried out the pathological examination. All authors have read and approved the final manuscript.


PEComa is a family of rare mesenchymal tumours which can occur in any part of the human body. Primary PEComas of the pancreas are extremely rare tumours with uncertain malignant potential. A 17-year-old female was admitted to the hospital due to melena. She required several transfusions. CT scan demonstrated a mass at the head of the pancreas measuring 4.2 cm in maximum diameter. An endoscopic ultrasound showed an ulcerating malignant looking mass infiltrating 50% of the wall of the second part of the duodenum in the region of the ampulla. Multiple biopsies taken showed extensive ulceration with granulation tissue formation and underlying large macrophages without being able to establish a definite diagnosis. We proceeded with pylorus-preserving pancreaticoduodenectomy. The postoperative course of the patient was unremarkable, and she was discharged on the 8th postoperative day. Histology examination of the specimen showed a PEComa of pancreas. Eighteen months after resection the patient is disease free. To the best of our knowledge this is the first time we describe a case of a pancreatic PEComa presenting with massive gastrointestinal bleeding. PEComa (perivascular epithelioid cell tumour) is a family of mesenchymal tumours consisting of perivascular epithelioid cells (PECs). PEComas are rare tumours that can occur in any part of the human body . The mosPrimary PEComas of the pancreas are extremely rare tumors with uncertain malignant potential. Only twelve cases, including the one we report here, are published in the literature , 4–13. SHere, we present a rare case of a patient with upper gastrointestinal bleeding due to an ulcerating head of pancreas PEComa. This patient underwent PPPD and 18 months after operation is disease free. To the best of our knowledge this is the first report of PEComa of pancreas manifesting with upper gastrointestinal bleeding.A 17-year-old female patient was referred to RMH due to melena caused by a mass at the head of pancreas. She presented at the local hospital 2 months before with melena. At that time she required several transfusions due to anemia (hemoglobin 6 g/dL at presentation) and she underwent oesophagogastroduodenoscopy (OGD), colonoscopy, and Meckel's scan; all of them reported as normal. A CT scan revealed a mass at the head of pancreas .At the time of referral she was asymptomatic. Her past medical history and the clinical examination were unremarkable. The review of the CT scan, which took place at the local hospital, demonstrated a lesion mass at the head of the pancreas measuring 4.2 cm in maximum axial diameter and 4.9 cm in the craniocaudal direction. This mass showed avid arterial phase enhancement with rapid washout, while it appeared almost isodense compared to the rest of the pancreas in the portal venous phase. Both pancreatic duct and common bile duct were prominent, with their diameter to upper normal limit. The SMV was abutment but not involvement. Extrapancreatic disease was excluded. These features were consistent with neoplasmatic mass of the head of pancreas, with the most possible pathology being a neuroendocrine tumour. The subsequent gut hormone test was normal .An endoscopic ultrasound (EUS) was performed showing an ulcerating malignant looking mass infiltrating 50% of the wall of the second part of the duodenum in the region of the ampulla. Multiple biopsies taken showed extensive ulceration with granulation tissue formation and underlying large macrophages without being able to establish a definite diagnosis.We proceeded with pylorus-preserving pancreaticoduodenectomy. The postoperative course of the patient was unremarkable, and she was discharged on the 8th postoperative day.Histology examination of the specimen showed an ulcerated tumour that had an expansible margin surrounded by a fibrous pseudocapsule. The tumor was well vascularised and composed of large mainly epithelioid cells with clear granular or feathery cytoplasm. Some cells with more spindle appearance were seen. The nuclei were eccentric, and there were many vascular spaces within the tumor which were dilated and some had irregular outlines. Mitosis was infrequent and there was no necrosis. No vascular invasion was seen. The cells were positive stained for HMB45, Melan A and smooth muscle actin. They were negative for cytokeratin, chromogranin, CD56, S100, desmin, and calponin. The above features were consistent with a perivascular epithelioid cell neoplasm (PEComa). Peripancreatic lymph nodes were negative for tumor and the resection was complete. Eighteen months after resection the patient is disease free.The perivascular epithelioid cell was first described in 1943 by Apitz as an “abnormal myoblast” in renal angiomyolipoma . Since tPEComas are well-circumscribed and set apart from the surrounding parenchyma by a thin capsule . HistopaFor the most part, PEComas are considered benign; however, a subset of PEComas behaves in a malignant fashion, leading to local invasion, multiple metastases, and death as observed with high-grade sarcomas. Recently, Folpe and Kwiatkowski have suggested criteria for malignancy, including a size greater than 5 cm, mitotic count of more than 1 per 50 high-power fields, and necrosis . PrimaryOur patient had a relatively small tumor . Mitosis was rare and no necrosis was seen; thus Folpe's criteria for malignancy are negative for our patient. Most PEComas present with abdominal pain but our patient's first symptom was melena.Radiographics findings with CT and MRI show that these lesions are significantly and heterogeneously enhanced on arterial phase, less enhanced on portal venous phase, and slightly hypodense on delayed phase . Early rSurgical resection represents the only curative approach for primary PEComa at presentation as well as for local recurrences and metastasis, as chemotherapy and radiotherapy have not demonstrated significant benefits . Only rePancreatic PEComas are rare entities which most commonly present with abdominal pain. However, this case shows that these tumours can manifest with upper gastrointestinal bleeding.


Actinomycosis is a rare chronic bacterial infectious disease in childhood. A 14-year-old boy admitted with cramping abdominal pain and vomiting. Physical examination revealed right lower quadrant tenderness. Appendectomy was performed. On the histological section, typical actinomycotic (sulfur) granules in the appendiceal lumen were observed. Actinomycosis is a rare chronic bacterial infectious disease in children and is characterized by multiple abscesses, fistulae, draining sinuses, granulation, and fibrous tissue. It is caused by Actinomyces species which was first described in 1878 by Israel. [1]Actinomycetes are filamentous, anaerobic, gram positive bacteria and are commensals of the oral and cervicofacial regions, respiratory tract,female genital organ, and gastrointestinal system.[2]In children, appendiceal actinomycosis with acute, perforated appendicitis is quite rare.[3]A 14-year-old boy was admitted with cramping abdominal pain and vomiting. Physical examination revealed right lower quadrant rebound tenderness. The white blood cell was 10,400/ul and serum C-reactive protein was increased at 3.56mg/dl. Increased (10mm) thickness of the appendix with nonperistaltic ileocecal region was noted by abdominal ultrasound (US). After preoperative preperation, appendectomy was performed. Grossly inflammed and enlarged appendix was noted. The pathologic specimen consisted of a vermiform appendix measuring 8cm x 3.5cm in dimension. The lumen contained a fecalith. The sections revealed acute inflamation. The inflammatory reaction was predominantly neutrophilic. With higher magnification typical actinomycotic (sulfur) granules in the appendiceal lumen were observed which were confirmed as appendiceal actinomycosis . Stains for other organisms including acid-fast basilli and fungi were negative. The appendix wall was in suppurative stage. The patient did well postoperatively.Actinomycosis is an uncommon disease.Actinomyces israelii is the most common cause of the Actinomycosis in humans.[4] Preoperative diagnosis of the Actinomycosis is a rare and difficult. In abdominal actinomycosis, the appendix and ileocecal regions are usually involved. Factors that predispose to abdominal actinomycosis include abdominal surgery, trauma, neoplasia, foreign bodies, and bowel perforation. Other predisposing factors are immunsuppression, malnutrition, and human immunodeficiency virus infections. Appendiceal actinomycosis is however rare in childtren. Abdominal actinomycosis may lead to nonspecific findings including mass, abscesses and sinuses and may be confused with diseases like diverticulitis, Crohn’s disease, ulcerative colitis and tubo-ovarian abscesses. The extensive fibrosis of actinomycotic lesions that appear as solid nodular or mass lesions are indistinguishable from malignancies. Sinus formation involving the perianal region can mimic Crohn's disease or tuberculosis. There are no spesific radiological features for actinomycosis. Hematological and biochemical markers are also nonspecific.The infection progresses insidiously and spreads along contiguous organs, especially the liver and may even involve the retroperitoneal tissues, spine and the abdominal wall. It rarely spreads by hematogenous or lymphatic dissemination. In our patient, actinomycosis was limited to the appendix with no evidence of distant organ involvement.[3]Antibiotic treatment consists of high doses of penicilin for four weeks. Treatment is continued for one year. Alternative first-line antibiotics include amoxicillin, tetracycline, erythomycin, and clindamycin.[4]To conclude, appendiceal actnimycosis is a rare pathology in children and should be listed in etiology of appendicitis.Source of Support: NilConflict of Interest: None declared


Morel-Lavallée lesions are cystic lesions occurring between the subcutaneous tissue and the underlying layer of a fascia. The most frequent mechanism of occurrence is posttraumatic, usually after degloving injuries. The chain of events leading to the occurrence of this lesion is the formation of a potential space between the subcutaneous tissue and the firmly attached deep fascia, which in turn fills with blood and/or lymph and/or necrotic fat, secondary to disruption of the capillaries. We present a case of a 74-year-old male patient with a cystic lesion of the lateral surface of his left thigh increasing in size over a period of six months. Despite the meticulous history acquisition, we were unable to reveal any alleged mechanism of injury of the area. The patient underwent an MRI which revealed an ovoid cyst. The patient underwent surgical excision of the cyst. The pathology examination revealed a simple cyst, lined by a fibrous capsule and filled with serosanguineous fluid, thus confirming the diagnosis. The patient made a full recovery and since has been asymptomatic. Morel-Lavallée lesion refers to a benign cystic lesion occurring within a potential space between the subcutaneous tissue and the underlying fascia , 2. MoreA 74-year-old male patient, with a past medical history of hypertension and chronic pain of his left hip joint, presented complaining of a cystic lump over his left hip joint which had increased in size during the past 6 months. The aforementioned lump was painless and was not associated with the movements of the hip joint. The patient did not report any recent medical history of injury of the associated area. Given his past medical history of chronic pain of his left hip joint, the patient underwent an MRI of the pelvis and thighs, in order to exclude a synovial cyst originating from the hip joint, secondary to chronic inflammation of the articular surfaces. The MRI revealed an ovoid cystic formation measuring 6.8 × 3.5 × 4.5 cm, with strong intensity signal on T1 and low on T2, lateral to the greater trochanter and the iliotibial tract . No pathMorel-Lavallée lesion refers to a cystic lesion secondary to blunt injury and especially in degloving injuries. When tangential shear forces are exerted on the relatively mobile subcutaneous tissue, due to inertia, the subcutaneous tissue is torn away from the relatively firm underlying muscle fascia. This results in the creation of a potential space between the two associated planes, which in turn is filled with blood/and or lymph from the disrupted perforating vessels/capillaries . Morel-LThe natural history of this lesion has not yet been established as it represents a rare entity. However some key steps of the process have been identified; after the initial formation of the potential blood filled space, there is evolution of this haematoma with absorption of the blood, which is replaced by serosanguineous fluid . The las


RECIST criteria. Surgical excision of the mediastinal mass was performed, and further postoperative treatment with adjuvant chemoradiotherapy was provided. Patient currently is free of disease. This is to the best of our knowledge the first report in the world literature of a successfully treated case of “primary mediastinal sarcomas presenting as SVC syndrome.” Patient is under regular surveillance at our clinic and remains free of recurrence one year after treatment completion.Primary mediastinal sarcomas are aggressive tumors with a very rare incidence. This report describes the case of a 35-year-old male patient who presented with acute symptoms of dyspnoea, facial puffiness, voice-hoarseness, and engorged neck veins. With the clinical picture consistent with the superior vena cava (SVC) syndrome, the patient was investigated with computed tomography of the chest. This revealed a large soft tissue density mass lesion compressing the SVC along with other critical superior mediastinal structures. Histopathological evaluation of the mass revealed features consistent with a soft tissue sarcoma and positive staining was observed for vimentin and S-100. Cytogenetic analysis by fluorescent in situ hybridisation (FISH) demonstrated the t(X:18) translocation. Thus diagnosis was established as primary mediastinal synovial sarcoma. Patient was treated with three cycles of neoadjuvant chemotherapy, to which there was a partial response as per the  Soft tissue sarcomas (STS) are rare malignant tumors comprising less than 1% of all malignant neoplasm. Among thoracic malignancies, the majority happen to be carcinomas, and the proportion of STS among thoracic malignancies is estimated to be as low as 0.01% –7.The current case report is possibly the first in the world of a patient with SVC obstruction due to a mediastinal synovial sarcoma who responded successfully to treatment with a combined modality approach utilizing neoadjuvant chemotherapy, surgery, postoperative chemotherapy, and radiotherapy. The patient has been rendered free of disease, currently at one-year follow-up after treatment completion.A 35-year-old nonsmoker male patient presented to the outpatient department with symptoms of rapidly progressive dyspnoea, facial puffiness, and voice-hoarseness developing over a span of 15 days prior to presentation. There was no history of any haemoptysis, abnormal swelling, breathing difficulty, headache, dizziness, tinnitus, epistaxis, or tuberculosis in the past. His overall past medical and surgical history was uneventful.On examination, the patient was tachypneic with stable vitals. There was a characteristic plethoric face with periorbital and upper body edema with dilated veins over the anterior wall of the chest and neck. There was no pallor, clubbing, lymphadenopathy, ptosis, or any signs of Horner's syndrome. On thoracic auscultation, there were decreased breath sounds in the third and fourth intercostal spaces in the right parasternal area and infraclavicular areas. The rest of the systemic examination was within normal limits.3, and platelet counts of 3,20,000/mm3. His serum electrolytes, renal function tests, and liver function tests were within normal range. Germ cell tumor markers like lactate-dehydrogenase (LDH), alpha-fetoprotein (AFP), and β-human chorionic gonadotropin (β-HCG) levels were within normal limits. Bone marrow examination did not show any malignant marrow infiltration. Serology was negative for human immunodeficiency virus (HIV), viral hepatitis B and C.His blood investigations at presentation revealed hemoglobin level of 11.5 gm/dL, total leukocyte counts of 8500 cells/mmContrast-enhanced computed tomography (CT) of the thorax showed an enhancing well-defined soft tissue density lesion of size 10.3 × 9.3 cm involving superior mediastinum, with compression of trachea, SVC, right upper lobe bronchus, and its branches . The masHistopathological examination of the biopsy specimen was characterized by a monotonous proliferation of tumor cells with oval to spindle, vesicular nuclei surrounded by an indistinct rim of amphophilic to lightly eosinophilic cytoplasm . Immunoh2 on days 1–5 and doxorubicin 37.5 mg/m2 on days 1 and 2) were given. Patient tolerated chemotherapy well, and there was partial response to the above mentioned chemotherapeutic drugs according to the RECIST criteria. Surgical excision of the mediastinal mass was carried out via a median sternotomy incision. Postoperative recovery was uneventful.Patient was initially managed symptomatically with diuretics, dexamethasone, oxygen inhalation, and propped up position for symptomatic superior vena cava syndrome. Palliative radiotherapy was omitted owing to the radioresistance of sarcomas. Three cycles of neoadjuvant chemotherapy . Patient was managed postoperatively with adjuvant postoperative radiotherapy (50 Gray in 25 fractions over 5 weeks), followed by three more cycles of chemotherapy. After completion of treatment, the patient is under regular surveillance and for the past one year remains free of recurrence.The term synovial sarcoma was earlier coined for the tumors arising near the joints. “Synovial sarcoma” owes the name to its resemblance to developing synovial tissue when seen under light microscopy. It however does not arise from the synovial tissue but arises from the pluripotential mesenchymal cells near joint surfaces, tendons, tendon sheaths, juxta-articular membranes, and fascial aponeurosis. These tumors comprise up to 5–10% of all soft tissue sarcomas. The most common age group of occurrence is in the second to fourth decades of life. The most common site of occurrence of these tumors is near the joints and lower extremities. The other sites that may be involved may be the head, neck, trunk, esophagus, intestine, mediastinum, and retroperitoneum. Primary mediastinal synovial sarcoma is a rare malignancy with only a few cases reported so far –7.The differential diagnoses for mediastinal synovial sarcomas would include other rare tumors such as malignant peripheral nerve sheath tumor (MPNST), thymoma, mesothelioma, pulmonary blastoma, and sarcomatoid carcinoma .Since diagnosis based on histopathological appearance in itself is difficult, it is imperative that additional methods such as immunohistochemistry and fluorescent in situ hybridization are necessary. Synovial sarcomas are usually positive for vimentin, epithelial membrane antigen (EMA), bcl-2, CD99 (60–90%), and S-100 (30%). Negativity for myogenin and myoD1 expression is seen, and these markers differentiate synovial sarcomas from rhabdomyosarcomas. Further, the presence of the translocation t(X:18) on fluorescent in situ hybridization is also confirmatory of synovial sarcoma .There are three subtypes of synovial sarcoma: the monophasic form , poorly differentiated form, and biphasic form. Monophasic type may be misdiagnosed as leiomyosarcoma, fibrosarcoma, hemangiopericytoma, and malignant peripheral nerve sheath tumor; thus the use of immunohistochemistry will be very much required to confirm a diagnosis. Poorly differentiated variant of synovial sarcoma can be diagnosed on the basis of expression of CD56 and CD59. Biphasic form is histologically composed of two sharply contrasted types of tissues: one of synovial element, the other of fibromatous element. Hence, the biphasic synovial sarcoma can be very easily identified based on characteristic histopathological findings alone . SYT located on chromosome 18q11 and SSX 1, SSX2 or SSX 4 located on Xp11 breakpoint [Thus, adequate tissue biopsy will be required for definitive diagnosis based on morphology, special staining, and chromosomal studies. Usually 90% of the synovial sarcomas demonstrate translocation involving fusion of two geneseakpoint .Synovial sarcomas traditionally have been regarded as being highly aggressive. The most common site of metastasis is lung; however other sites that are less commonly involved include lymph nodes, bone, and bone marrow. Important factors of prognostic significance include the presence of distant metastasis, tumour size (>5 cm), resection margin status, and degree of histological differentiation. Achieving negative surgical margins is highly important, and in case of gross residual disease, reexcision should always be considered. Radiotherapy and adjuvant chemotherapy can enhance the likelihood of disease control.When complete surgical resection is not possible due to factors such as proximity of tumor to critical structures, maximum possible resection followed by adjuvant radiotherapy is the favored treatment . SynoviaPatients with unresectable localized disease can be managed with the aim of curing by the use of neoadjuvant chemotherapy and/or radiotherapy so as to obtain a regression which may allow subsequent surgical resection . In our


The current report presents the case of a 43-year-old male suffering from a penile cutaneous horn. A surgical excision of the lesion was performed and histopathology demonstrated hyperkeratosis, dyskeratosis and epithelial hyperplasia. The cutaneous horn progressed to squamous cell carcinoma <1.5 months following surgery and a partial penectomy was conducted. The International Index of Erectile Function 5 questionnaire was used to assess the patient and the score had decreased in the one-month postoperative follow-up compared with that of the preoperative period. These findings indicate that undergoing a partial penectomy on initial diagnosis of a penile cutaneous horn should be considered in order to conserve a greater quantity of the penile tissue and improve the postoperative quality of life. The incidence of penile cutaneous horn has been particularly low since the first case was reported . It is aIn February 2013, a 43-year-old male presented to the Department of Urology, The Third Xiangya Hospital of Central South University  with a six-month history of a progressive growth in the glans penis. The patient had undergone a circumcision for phimosis six months previously and subsequently identified a yellow lesion projecting out from the left aspect of the glans penis. The patient experienced no pain and disregarded the lesion, which grew rapidly and prevented the patient from engaging in sexual intercourse. Examination of the patient revealed a horn-like keratinized lesion of ~2.0×1.0×0.6 cm in the glans penis with no inguinal lymphadenopathy . The IntThe first postoperative visit was conducted on day 39 following surgery, the patient presented with a painful swelling over the glans penis and purulent discharge of the urethra. On examination, the glans penis was stiff and there was a ~5-mm nodular lesion on the distal aspect of the penile shaft. Pelvic magnetic resonance imaging (MRI) revealed a lesion with an unclear boundary measuring 0.6 cm in diameter . A biopset al (in situ DNA hybridization; furthermore, Zhu et al (The etiology of penile cutaneous horn remains uncertain since the first case was reported in 1854 (hu et al ,7. To thhu et al . HoweverThe European Association of Urology guidelines on penile cancer classify penile cutaneous horn as a premalignant lesion  and apprAs instances of penile cutaneous horn are particularly rare and the majority of studies regarding them are case reports, various treatment methods have been adopted, including laser therapy, administration of keratolytic agents and surgical excision ,11. In tIn conclusion, to the best of our knowledge, such a rapid progression of a penile cutaneous horn to squamous cell carcinoma has not previously been described. In addition, a decline in the patient’s quality of life was noted. Thus, performance of a partial penectomy should be considered as soon as a diagnosis of penile cutaneous horn is determined.


Osteochondroma is rarely reported in the maxillofacial region; however, it is prevalent in the mandibular condyle. This slowly growing tumor may lead to malocclusion and facial asymmetry. A 39-year-old woman complained of gradual development of anterior and posterior unilateral crossbite, which resulted in facial asymmetry. A radiological study disclosed a large tumor mass on the top of the left mandibular condyle. This bony tumor was surgically removed through condylectomy and the remaining condyle head was secured. Subsequently, bimaxillary orthognathic surgery was performed to correct facial asymmetry and malocclusion. Pathological diagnosis was osteochondroma; immunohistochemistry showed that the tumor exhibited a conspicuous expression of BMP-4 and BMP-2 but rarely expression of PCNA. There was no recurrence at least for 1 year after the operation. Patient’s functional and esthetic rehabilitation was uneventful. Osteochondroma is one of the most common benign bone tumors  . HoweverMany options can be considered for the treatment of osteochondroma, including resection via local excision (condylectomy), arthroplasty, and vertical ramus osteotomy. Reconstruction with an autogenous bone graft such as costochondral graft or total joint replacement with a Temporomandibular joint prosthesis can also be good treatment options .If the patient has malocclusion, two-step approaches, such as resection followed by orthognathic surgery have been used. However, there are few reports of mass resection with simultaneous orthognathic surgery. Here, we describe a case of mandibular condyle osteochondroma treated with bimaxillary orthognathic surgery as well as condylectomy.A 39-year-old woman was referred for facial asymmetry and malocclusion, which had slowly progressed over 4 years. She visited a dental hospital 2 years before admission and was diagnosed with chondroma by radiological observation. She did not experience any systemic diseases or accidental trauma. Although she had been treated for malocclusion in a local clinic, her malocclusion was not appropriately corrected but gradually worsened. Written informed consent was obtained from the patient for the publication of this report and any accompanying images.Clinical examination revealed severe malocclusion and facial asymmetry. Intraorally, her midline of the mandibular teeth was deviated to the right side by up to 12 mm , which was diagnosed as probable osteochondroma. In cone-beam computed tomography (CBCT), bone marrow and a bony trabecular pattern were observed .Since the patient’s chief complains were facial asymmetry and malocclusion, condylectomy on the left condyle with simultaneous bimaxillary orthognathic surgery was planned. Because of the tumor growth, the patient’s maxillary dental arch was canted right and up by 3 mm in cephalometric analysis. Under general anesthesia, Le Fort I osteotomy was performed to correct maxillary canting. Sagittal split ramus osteotomy was also performed on the right side of the mandible and vertical ramus osteotomy was performed on the left side by a submandibular approach. After taking out a condylar segment, the irregularly out-growing tumor was removed along with a portion of normal tissue and condylectomy was also performed. To level both TMJs, a 5-mm fragment of cortical bone was harvested from the exposed mandibular angle area and grafted below the osteotomized segment Figure . Then, tOn histological examination, a chondroid mass was found on the capsule of the mandibular condyle, which was extended into underlying trabecular bones. The chondroid tissue showed many hyperplastic chondrocytes, which were mostly surrounded by hyalinized matrix and subsequently underwent ossification to produce trabecular bones. The underlying trabecular bones were irregular in shape and anastomosed each other, resulting in cancellous bone with abundant marrow stromal tissue Figures ; A1, A2.The post-operative course was uneventful. Intermaxillary fixation was performed to stabilize the jaws for 2 weeks. The patient performed jaw movement exercises for 3 months after removal of intermaxillary fixation. At 12 months post-operation, the range of maximum mouth opening was 40 mm without pain or any interference, and no signs of recurrence were observed Figures .Figure 4Osteochondroma is a benign tumor of bone and cartilage. It is common in axial skeleton bones such as femur or tibia, but rare in the maxillofacial region . In the A genetic component may be involved in this disease . AnotherSome patients exhibit vertical elongation of the affected side and slight pain . PanoramFacial asymmetry and malocclusion may also be observed in condylar hyperplasia and other differential diagnoses such as osteoma, chondroma, fibrous dysplasia, fibrosarcoma, and chondrosarcoma . Thus, hOn histological observation, the present osteochondroma showed diffuse proliferation of chondroid tissue, which partly produced ossifying trabecular bones. The chondroid tissue was conspicuously positive for BMP-4 and the trabecular bones were slightly positive for BMP-2. Most chondrocytes were surrounded by hyalinized chondroid material and showed rare PCNA immunoreaction. Therefore, we presume that the present tumor was derived from condyle chondrocytes that showed ossification, and are confident in the osteochondroma diagnosis. We also believe that the present osteochondroma was a relatively well-differentiated benign tumor with low proliferative potential.The protocol for treatment of osteochondroma of the mandibular condyle is controversial. If only the head of the condyle is involved without tumor extension into the neck, local resection or conservative condylectomy with contouring the affected condylar head can be the appropriate choice . HoweverDeviation of the mandible because of osteochondroma of the mandibular condyle can also change the occlusion plane. In this case, orthognathic surgery should be considered. It can re-establish optimal occlusion and improve facial aesthetics . There aOsteochondromas of the mandibular condyle can represent facial asymmetry, malocclusion, and TMJ pain. It can also occur occlusion plane disharmony between maxilla and mandible. Bimaxillary orthognathic surgery is able to improve the occlusion status as well as the esthetic facial profile, simultaneously. The condylectomy with orthognathic surgery can be good choice to treat osteochondroma of mandibular condyle in facial deformity patients.


Tension pneumoperitoneum is a rare surgical emergency in which free intraperitoneal gas accumulates under pressure. The known sources of free gas are perforated hollow viscera. We believe this is the first published case of a tension non-perforation pneumoperitoneum secondary to anaerobic gas production. This occurred in a background of primary non-aerobic bacterial peritonitis, which developed in an immunocompetent adult man.A previously healthy 45-year-old Bulgarian man presented with a 3-week history of abdominal pain. He displayed signs of shock, peritonitis, and abdominal compartment syndrome. A plain abdominal X-ray showed the pathognomonic “saddlebag sign” with his liver displaced downwards and medially. An emergency laparotomy released pressurized gas, accompanied by 3100 mL of foamy pus. A sudden hemodynamic deterioration occurred soon after decompression. The sources of infection and tension pneumoperitoneum were not found. The peritoneal exudate sample did not recover aerobes. A laparostomy was created and three planned re-operations were performed. During the second re-laparotomy we placed an intraperitoneal silo and his abdomen was closed with skin sutures. Definitive fascial closure was achieved through separation of his two rectus muscles from their posterior sheaths. He was discharged in good health on the 25th postoperative day.Our case provides evidence supporting the theory that anaerobic infection may underlie the etiology of tension pneumoperitoneum. Prior to decompressive laparotomy the patient should receive an intravenous volume bolus to compensate for possible hypotension. If laparostomy leads to lateralization of the rectus muscles with a gap of 6 cm or less, the posterior part of the components separation technique is effective in achieving fascial closure. We present an original classification of tension pneumoperitoneum defining it as primary or secondary.The online version of this article (doi:10.1186/s13256-016-0945-0) contains supplementary material, which is available to authorized users. Tension pneumoperitoneum (TP) is the accumulation of free gas under pressure within the peritoneal cavity leading to abdominal compartment syndrome (ACS). The latter is manifested by respiratory failure, which is due to compression of the diaphragm, and obstructive shock, which is the result of compression of the intra-abdominal veins. Compensatory peripheral vasoconstriction can maintain perfusion of vital organs for a limited time. It is pertinent that this situation may be a precursor to an unexpected cardiorespiratory arrest. The diagnosis is based on the clinical signs and symptoms, supported by a plain abdominal X-ray. Radiological findings include pneumoperitoneum, diaphragmatic elevation, downward and medial displacement of the liver (saddlebag sign), with centralization of bowel loops . If TP oSeven electronic libraries were browsed to identify English language reports on TP. The period was from 1919  until 2015. The search revealed 124 publications with 159 cases of TP. A reference of significance was made by Singer to three German authors 1913 to 1914) who hypothesized that free gas accumulation could be due to gas-producing microorganisms [ to 1914 Our review revealed that all reported cases were secondary to perforations of hollow viscera. The digestive and respiratory systems were primary locations, with one case originating from the bladder. Of the 159 cases, 127 occurrences of TP were secondary to gastrointestinal perforations caused by: iatrogenic injury, 64; disease, 42; barotrauma and trauma, 11; and surgery, 10. Thirty one cases followed respiratory perforations caused by: iatrogenic injury, 4; barotrauma, 20; blunt chest trauma, 6; and surgery, 1. One case followed a bladder perforation caused by a Foley catheter left open to air. This analysis revealed that the incidence of TP resulting from gastrointestinal perforations was four times higher than those caused by ruptured respiratory organs  measured via urethral catheter was 26 mmHg. This was a grade IV intra-abdominal hypertension (IAH). The amount of urine on catheterization was 50 mL. Blood tests showed anemia, leucocytosis, azotemia, hypoproteinemia, and metabolic acidosis; alpha-amylase was normal. An erect plain X-ray of his abdomen revealed the saddlebag sign Fig. . A chestHe was in shock, with signs of generalized peritonitis and ACS. He received a short course of intravenous fluid resuscitation and broad-spectrum antibiotics. This was immediately followed by a midline laparotomy. Upon entering his peritoneal cavity, pressurized gas with a “rotten-egg” odor escaped and 3100 mL of frothy fetid pus evacuated. Despite preoperative rehydration, decompression of the abdomen resulted in sudden hypotension.All of his organs and tissues were uniformly inflamed with no apparent focal site. His intestines were gathered medially in a ball and covered by a “shield” of dense fibrin deposits below which they appeared normal with shiny serosa and non-swollen walls. The appendicular end was indistinguishable from his small bowel loops, and beneath the fibrin shield looked normal. An appendectomy was performed.Systematic exploration of all intraperitoneal, retroperitoneal organs and spaces (including the lesser sac) did not reveal the source of infection. Air-leak testing excluded a ruptured hollow viscus. A sealed perforation was also ruled out. Therefore we concluded that we were dealing with a primary bacterial peritonitis (PBP) of an anaerobic microorganism whose metabolism was responsible for the TP.His abdomen was washed out and left open. His IAP was monitored during the first 48 hours, and fluctuated between 10 and 15 mmHg. Three planned re-laparotomies were performed at 48-hour intervals for debridement, wash out, and exploration for any septic source; none were found. At the end of each re-entry, the laparostomy was partially closed with fascial sutures at both ends. Rapid degradation of the fibrin shield made the bowel loops distinguishable on the second re-entry Fig. . On thiset al. [On the third re-laparotomy, the silo was removed and the decision was made to definitively close his abdomen. Fascial suture was not possible because the central section remained with an insurmountable gap of 6 cm. We performed the posterior part of the components separation technique (CST) described by Ramirez et al. . LongituAcinetobacter baumannii (anitratus). Blood samples were sterile. This bacterium is a Gram-negative obligate aerobe that usually causes nosocomial infections that are rarely clinically significant. With sensitivity only to carbapenems, a 6-day course of imipenem (1.5 g/day) led to complete eradication. The exudate samples for biochemical testing for bilirubin and alpha-amylase revealed levels corresponding to non-elevated serum samples.The sample of the peritoneal exudate for culture and sensitivity taken during the initial operation did not grow bacteria. The samples from the three re-operations as well as the urine culture recovered A histological examination of the removed appendix showed that its end was reactively inflamed from the outside in. The mucosa was intact, as was the proximal appendicular wall through its full thickness. This excluded the appendix as the primary source of the infection.Our patient was negative for HIV, hepatitis B, and hepatitis C. He had normal levels of immunoglobulins and was immunocompetent. Before discharge, the following imaging tests were performed: (a) a follow-through with liquid contrast, which revealed no structural or transit-times abnormalities throughout his gastrointestinal tract; (b) a computed tomography of his abdomen, which showed normal findings for all parenchymal and hollow organs.He was discharged in good health 25 days after admission. He had no complaints at the 1- month and 6-month follow-up. He gained 10 kg in weight and returned to work. His abdominal wall was healthy; an abdominal ultrasound and chest X-ray were normal. A tuberculosis screening test was negative. He continued to be symptom-free with normal findings on physical examination 18 months after discharge.The main characteristic aspect of the TP in our case was its slow development. This avoided the need for abdominal paracentesis and determined the clinical presentation. The slow progression of the disease allowed the accumulation and subsequent compression of free gas from anaerobic respiration. In addition, this enabled the mechanical processes of the TP to overlap the immune responses to peritonitis. This gradual increase in IAP provided time for the respiratory and cardiovascular compensatory reserves to intervene. Consequently, the clinical signs were less dramatic compared to cases of TP described in the literature search.Normally, decompression of the abdomen results in rapid hemodynamic stabilization. Although rare, abdominal decompression can cause further hemodynamic deterioration. An immediate drop in IAH can “steal” blood flow, diverting it to the splanchnic area, and reducing heart preload to critical levels. This effect and concomitant decrease in systemic vascular resistance result in a sudden drop in aortic pressure. Reperfusion injury may occur and lead to extreme hyperkalemia and possible cardiac arrest. Therefore, shortly before decompression, the patient should receive an intravenous volume bolus . If hypoThe lack of bacterial growth in an apparent infectious peritonitis without previous antibiotic therapy suggests that an obligate anaerobic bacterium is the sole causative agent. It could not be a facultative anaerobic bacterium or a mixed infection because they would culture on standard agar medium in the presence of oxygen .2S). This gas is one of the end products of anaerobic respiration and not fermentation. Fungi only use fermentation consequently they could not be the causative agent in this case. This also excludes other aerobes such as fastidious bacteria, mycobacteria, and viruses, none of these produce H2S. Note that certain aerobic sulfate-reducing bacteria such as Desulfovibrio organisms can produce H2S, but they would recover on standard agar medium [The odor of rotten eggs is caused by hydrogen sulfide .Although rare, TP is well documented but without classification. Based on the source of gas, we use and present our original classification Table :“PrimaryThe case here was placed in group (a).The combination of TP without perforated hollow viscus and primary anaerobic bacterial peritonitis is an extremely rare scenario in emergency surgery. Our case study provides evidence supporting the infectious etiology of TP. Shortly before laparotomy, the patient should be overhydrated to avoid decompression hypotension. The existing TP in a background of severe abdominal sepsis necessitates laparostomy. The posterior part of the Ramirez operation can be safely utilized, providing the insurmountable gap is ≤6 cm. We classify TP as primary or secondary.2S, hydrogen sulfide; HIV, human immunodeficiency virus; IAH, intra-abdominal hypertension; IAP, intra-abdominal pressure; PBP, primary bacterial peritonitis; RMFC, rectal myofascial complex; TP, tension pneumoperitoneum.ACS, abdominal compartment syndrome; CST, components separation technique; H


Intraosseous hemangiomas account for 1% of all bone tumors and primarily originate from the vertebral column and skull bones. However, intraosseous hemangiomas of the nasal cavity are extremely rare. Here, we report a case of intraosseous hemangioma with a cavernous pattern arising from the middle turbinate that was preoperatively misdiagnosed as chronic rhinosinusitis with polyps. Except for nasal obstruction, there were no specific rhinologic symptoms. The tumor was excised en bloc by the endoscopic endonasal approach without preoperative embolization. Tumors of the nasal cavity and paranasal sinus have different manifestations including hemangiomas. Hemangiomas are benign vascular tumors and account for about 20% of all benign nasal cavity tumors . HemangiA 64-year-old man with a long history of left nasal obstruction but no epistaxis or facial trauma was referred to our clinic. Anterior rhinoscopy revealed septal deviation to the right and a polyp-like mass occupying the left nasal cavity. We performed an endoscopic biopsy and enhanced computed tomography (CT) at the outpatient clinic. The histologic examination showed the polyp with fibrinoid necrosis . CT scanThese findings were suggestive of chronic rhinosinusitis combined with either nasal polyps or a benign tumor; therefore, we performed a left endoscopic sinus surgery along with mass excision under general anesthesia. Additionally, a partial middle turbinectomy was performed to remove the mass filling the bottom of the middle turbinate. The tumor was removed transorally after pushing it toward choana. During the surgery, the tumor (5 × 3 × 2 cm) was found to be covered with normal mucosa, with the whole sinus containing mucopurulent discharge and polypoid mucosa. There was no significant bleeding and the left nasal cavity was packed with nonabsorbable materials. The packing was removed on the second postoperative day, after which the patient was discharged home without any complications. The histologic examinations revealed that the tumor comprised anastomosing thin-walled vascular channels with a cavernous pattern between the bony trabeculae and the normal mucosa covering it . TherefoHemangiomas are histopathologically classified into 3 types: capillary, cavernous, and mixed. The capillary and cavernous types are divided according to the prominent vessel size. The mixed type hemangiomas comprise a proliferation of thin-walled blood vessels of several sizes lined by endothelium .Hemangiomas of the head and neck are common but those of the nasal cavity and paranasal sinuses are rare. The most common site for nasal hemangiomas is the nasal septum followed by the lateral wall and vestibule , 4, 5, 8The pathogenesis of intraosseous hemangiomas remains unknown. Nonetheless, local trauma may be a cause, because many patients with intraosseous hemangiomas have had a history of local trauma , 3–5. ItCT is a useful imaging technique for the diagnosis of intraosseous hemangiomas, because of its ability to reveal the characteristic soap bubble or honeycomb patterns , 3–6. OtThe most effective treatment to prevent recurrence of intraosseous hemangiomas is complete surgical resection , 3–5. OtIn summary, intraosseous hemangioma of the nasal cavity is an extremely rare disease that is difficult to diagnose without a biopsy. Therefore, it may be necessary to consider a differential diagnosis for intraosseous hemangiomas in the presence of a polyp-like lesion. The typical histological and radiologic characteristics of intraosseous hemangiomas of the nasal cavity will be helpful to make the appropriate diagnosis and effectively manage the condition. Intraosseous hemangiomas arising from the middle turbinate can be excised completely through an endoscopic endonasal approach without the risk of any complications.


Epidermoid cysts are common cystic lesions in the skin, ovaries, and testicles, but their occurrence in the oral cavity is uncommon. They consist of cysts delimited by a fibrous capsule without cutaneous annexes and are lined by stratified squamous epithelium. The differential diagnosis includes ranula, dermoid cysts, and lingual thyroid. Despite their benign presentation, these cysts can cause functional limitations, requiring special clinical attention for extensive lesions located in regions that preserve vital structures. This paper aims to report a case of epidermoid cyst in patient with swallowing and breathing difficulty, highlighting the clinical and surgical planning. Epidermoid cysts  are common cystic lesions in skin, testicles, and ovaries derivates of ectoderm-lined inclusion. They comprise less than 0.01% of all oral cavity cysts and their occurrence in the floor of the mouth with respiratory complications has not been reported .Histologically, EC are bounded by fibrous capsule and are composed of an epithelium which is flattened and contains a granular layer of keratohyalin granules. Absence of hair follicles, sebaceous glands, and apocrine sweat glands (skin appendages) in capsule of these cysts helps differentiate between dermoid cysts . The derEtiology of EC remains unknown and the most accepted theory is the reactivation of the remaining ectoderm trapped in the 1st and 2nd pharyngeal arches. However, accidental or traumatic inclusion of epithelial tissue in deep structures of the dermis or submucosa may be associated with pathogenesis of epidermoid cysts , 6.The epithelial cells malignant transformation of these cysts has been reported but is rare , 8. ExteThis paper aims to report a case of an extensive epidermoid cyst on buccal floor, with emphasis on the importance of image diagnosis (Cone-Beam Computed Tomography) for treatment planning.A 45-year-old male patient presents with extensive mass in the buccal floor, with limitation in mouth opening and speech associated with dysphagia and dyspnea. The period of evolution of lesion was unknown. The clinical examination revealed an expansive mass, asymptomatic, exophytic, and no history of associated trauma, and fluctuated upon palpation . The lesAs patient reported swallowing and breathing difficulty, additional hematological examinations were performed, which showed normal range values. For surgery planning purposes and for observed relationship with soft tissues and other anatomical structures, a CT scan was performed showing the dimensions of the lesion, as well as confirming the hypothetical diagnosis of the internal liquid contents. Aspiration puncture demonstrated content material friable and white. Surgical planning included complete lesion excision. After this the specimens were removed and were sent for anatomical pathologic evaluation.The ovoid cystic mass was macroscopically observed to be opened and without any content. It was measured to be 5.0 × 3.0 × 0.2 cm and it had brown pigmentation with a few whitish areas. Microscopic examination revealed a cystic cavity with a capsule composed of dense fibrous connective tissue, lined by stratified squamous epithelium resembling epidermis . There wThe epidermoid cysts (EC) have uncertain etiology and may be formed from reactivation of epithelial remnants entrapped during midline closure of the bilateral first and second branchial arches. Another probable cause is accidental introduction of epithelium in the subcutaneous tissues or in the submucosa after extraction of a third molar, for example, , 11, 12.They affect mainly male patients and are uncommon during childhood or puberty. Their occurrence in the oral cavity is rare and represents less than 0.1% of all oral cysts , 13. DesEC have different diagnosis such as infectious lesions of the salivary glands, ranula, dermoid cyst, lipoma, lingual thyroid, and thyroglossal duct cyst , 14–16. Computed tomography (CT) is a reliable assessment of lesion extension to deeper structures when diagnosing or evaluating the submandibular space. CT permits visualization of the differences in densities of hard and soft tissues thus optimizing the diagnosis and it guides the surgeon for a more efficient treatment plan and enables visualizing the relationship of the lesion with muscles, salivary glands, and other tissues , 17, 18.In this case for safe surgical approach,was observed the depth of the lesion in the submandibular space. Also it became evident that the lumen of the lesion is of low density, assisting in the appropriate development of a clinical diagnostic. Expansion of the lesion was confirmed with a reduction of nasopharynx space, observed in CT images .The imaging examinations also allow selecting the surgical approach: intra- or extraoral. In our case we decided to use the intraoral approach, despite a large dimension cyst, which presented superficial involvement. During surgical procedures the contents of the cysts revealed friable and white material compatible with keratin .The dermoid cyst can occur in the skin, ovary, testicles, and other regions of the body always related to the midline, because it matches teratomatous injuries. In the capsule, microscopic examination showed the presence of skin appendages in the cyst wall, in addition to the stratified squamous lining. The gonadal lesions often exhibit other mature tissues such as cartilage, bone, fatty tissue, and nerve tissue, as well as skin structure . AccordiA total lesion excision was the recommended treatment, and the recurrence is unusual. The risk of malignancy of these cysts is rare, but there are reports on dental literature. The etiology and pathogenesis of the EC require more conclusive studies .As mentioned, epidermoid cysts are benign lesions; however, they may have large dimensions and cause physiological complications including swallowing and breathing difficulty. The computed tomography is a method efficient for assessing the relationship with the adjacent anatomical structures and planning of surgical approach.


A 38-year-old male was admitted to Renji Hospital  with the major complaint of back pain due to left hydronephrosis. Imaging analysis revealed an area of nodular soft-tissue density in the left ureteral wall. The patient’s left kidney was non-functional. Thus, a left nephroureterectomy was performed for the purpose of pathological diagnosis, and histopathological examination revealed follicular lymphoma. The patient received R-CHOP chemotherapy  every three weeks. Following six courses of chemotherapy, positron emission tomography-computed tomography revealed that the patient was in complete remission. From this case we showed that in cases where a partial ureteral stenosis with ureteral wall thickening was observed by imaging analysis, further histological examination of tissue samples should be assigned as soon as possible. Non-Hodgkin’s lymphoma (NHL) has a far greater inclination to disseminate to extranodal sites compared with Hodgkin’s lymphoma (HL). The most common extranodal site is the gastrointestinal tract, whereas NHL of the ureter is rare. Over the past five decades, there have been limited case reports regarding NHL of the ureter. Due to the limited number of cases, treatment of the disease has not been unified. In the current study, a Chinese male with NHL of the ureter was identified. The patient was treated with surgery and chemotherapy. In combination with the relevant literature, the diagnosis and treatment of malignant lymphomas of the ureter are discussed in the present study.In October 2011, a 38-year-old Chinese male complained of back pain and was admitted to Changzheng Hospital  with left hydronephrosis, which was confirmed by ultrasound. Ureteroscopy revealed a luminal stenosis of the left ureter, thus, a ureteral double J stent was inserted. A ureteroscopic biopsy was performed and histopathological examination revealed a granuloma. Follow-up examination months later showed no evidence of diminished hydronephrosis. Written informed patient consent was obtained from the patient.2, while the GFR of the right kidney had reduced to 38.3 ml/min/1.73 m2. Subsequently, the patient underwent a left nephroureterectomy. Pathological examination revealed that the left-middle ureter exhibited nodular thickening. Additional immunohistochemical analysis of the specimen  for further evaluation of the ureteral stenosis with uncertain etiology. Magnetic resonance imaging (MRI) revealed an area of nodular soft-tissue density in the wall of the left-middle ureter . This sespecimen  and 4 re2 rituximab, 750 mg/m2 cyclophosphamide, 20 mg/m2 liposomal doxorubicin, 1.4 mg/m2 vincristine and 80 mg methylprednisolone (R-CHOP), which was administered every three weeks. Following three courses of treatment, PET-CT examination revealed that the paraaortic adenopathy had reduced by 50%, the iliac nodes had disappeared and FDG metabolism in the sternum and right ilium was normal. Following six courses of chemotherapy, the PET-CT scans showed negative for residual tumor. Subsequently, the patient received a further two courses of R-CHOP. Thus far, the follow-up period is seven months and the current evaluation is that the patient is in complete remission.The patient received systemic chemotherapy combining 375 mg/mClinical manifestation of NHL is diverse and NHL has highly variable outcomes. NHL has a far greater inclination to disseminate to extranodal sites. The extranodal sites normally affected by NHL include the gastrointestinal tract, testes, ovaries, central nervous system, prostate, thyroid, bones and skin. The most common extranodal site is the gastrointestinal tract, whereas NHL of the ureter is rare. An extensive literature search of the PubMed database only identified a few case reports –3,5–9 ovet al (There is no particular imaging characteristic and in the majority of cases, diagnosis was established according to the histopathological study of tissue samples obtained from partial ureterectomies or nephroureterectomies. Hashimoto et al  reportedet al . Adventiet al (Overall, 25–40% of NHL patients present with a primary extranodal lymphoma. However, the definition of primary extranodal lymphoma is controversial, particularly in patients where nodal and extranodal sites are involved. Certain studies have indicated that only patients with localized nidus have primary extranodal lymphoma –13. Alteet al  hypothesIn conclusion, in cases where a partial ureteral stenosis with ureteral wall thickening is observed by imaging analysis, further histological examination of tissue samples should be assigned as soon as possible, however, tissue biopsy via ureteroscopy is not recommended.


Case Presentation. A woman on warfarin with SLE and the antiphospholipid syndrome, presented with left loin pain. She was normotensive and clinically normal. Ultrasound scans demonstrated left kidney scarring, but CT scans revealed an AI. MRI scans later confirmed the AI without significant fat and no interval growth. Cortisol after 1 mg dexamethasone, urinary free cortisol and catecholamines, plasma aldosterone renin ratio, and 17-hydroxyprogesterone were within the reference range. Initially, adrenal haemorrhage was diagnosed because of warfarin therapy and the acute presentation. However, she underwent adrenalectomy because of interval growth of the AI. Histology confirmed an RL. The patient received adjuvant radiotherapy. Discussion. Our subject presented with an NSAI. However, we highlight the following: (a) the diagnosis of adrenal haemorrhage in this anticoagulated woman was revised because of interval growth; (b) the tumour, an RL, was relatively small at diagnosis; (c) this subject has survived well over 60 months despite an RL perhaps because of her acute presentation and early diagnosis of a small localised tumour.Adrenal incidentalomas (AIs) are mostly benign and nonsecretory. Management algorithms lack sensitivity when assessing malignant potential, although functional status is easier to assess. We present a subject whose AI was a retroperitoneal leiomyosarcoma (RL).  Adrenal incidentalomas (AIs) are a modern phenomenon  and seveAIs are usually benign and nonsecretory . The curWe present a subject who had a very rare high-grade retroperitoneal leiomyosarcoma (RL) presenting as an NSAI. She also had the antiphospholipid syndrome and was on warfarin therapy.A 40-year-old woman with SLE and the antiphospholipid syndrome presented acutely with sudden onset left sided loin pain. She was on life-long warfarin therapy for multiple DVTs and pulmonary emboli and hydroxychloroquine. Ultrasound scans showed a 10 mm focal area in the left kidney due to scarring or a small angiomyolipoma. A CT scan several months later showed a 2.7 × 3.9 cm left adrenal incidentaloma , and sheOn examination, she was normotensive and had no clinical signs of endocrinopathy. Investigations including cortisol after 1 mg dexamethasone suppression overnight, urinary free cortisol (twice), urinary catecholamines (twice), aldosterone renin ratio, 17-hydroxyprogesterone, and chromogranins were all within the reference range. MRI scans showed a solid left adrenal lesion measuring 2.4 × 4 cm. without significant fat and no interval growth. However, in view of the phenotype and size of the adrenal mass she was referred to the endocrine surgeons. Following a review of her history of loin pain, and imaging, it was decided that she had a likely benign NSAI , who may have developed acute symptoms as a result of haemorrhage into the lesion. Therefore a conservative approach was adopted with scans to be repeated in 3–6 months. Although the size of the lesion initially remained stable, imaging one year later revealed that it had increased in size to 3 × 5.4 cm. The patient therefore had left laparoscopic adrenalectomy. At surgery, there was no macroscopic invasion of adjacent structures and the lesion was excised with periadrenal fat.Histology revealed a nonencapsulated high-grade leiomyosarcoma thought to have arisen from connective tissue but separate from the adrenal gland, that is, a retroperitoneal leiomyosarcoma (RL). There were atypical pleomorphic spindle cells forming a herringbone pattern in areas . These cWe have described a 40-year-old woman who presented with what appeared to be a benign NSAI. It was later confirmed that the “NSAI” was actually an RL and separate from the adrenal gland. As far as we are aware, this is the first report of an RL presenting as an AI. Retroperitoneal sarcomas are rare and RL even more so . They arThe management of our subject was complicated by her drug therapy and the nonspecific appearances on imaging. It is noteworthy that her initial abdominal ultrasound scan was reported to show renal scarring or an angiomyolipoma of the kidney, both benign entities. An AI was only identified later on CT scanning. Endocrine assessment at this stage confirmed it to be an NSAI, but despite a lack of interval growth, it was felt, on size criteria and a paucity of fat in the lesion on imaging, that surgery should be considered . The comThe diagnosis of an adrenal bleed in this anticoagulated woman was revised because of interval growth of the mass.This growth was only detectable 1 year after first evaluation. Lack of early interval growth does not exclude a malignant lesion in every subject; we support repeating initial negative scans as recommended in recent algorithms .Surgery should be considered for 4 cm diameter AIs showing an appropriate noncontrast CT phenotype.While primary adrenal leiomyosarcomas are very rare (only 20 cases reported so far), RL may very rarely present as an NSAI.This tumour was relatively small compared to typical RLs described in the literature—50% more than 15 cm in diameter. This was most likely because of her acute presentation with loin pain, which prompted imaging which led to an earlier diagnosis.This subject has survived for well over 60 months despite a high-grade malignant RL perhaps because of early detection and a small localised tumour, resulting from her acute presentation.In conclusion, we would like to highlight the following.


However, rare cases of false positive reactions have been reported. A 67-year-old male patient was admitted with epigastric pain. Abdominal computed tomography (CT) revealed a heterogeneous left adrenal mass 6 cm in diameter; following hormone testing, 123I-MIBG scintigraphy was performed to determine the presence of pheochromocytoma, which confirmed eccentric uptake by a large left adrenal gland mass. Chest CT and PET-CT confirmed metastatic lymphadenopathy; therefore, endobronchial ultrasound transbronchial needle aspiration was performed. Metastatic carcinoma of unknown origin was suspected from a lymph node biopsy, and surgical resection was performed for definitive diagnosis and correction of excess hormonal secretion. A final diagnosis of undifferentiated adrenal malignant tumor was rendered, instead of histologically malignant pheochromocytoma, despite the uptake of 123I-MIBG demonstrated by scintigraphy. Pheochr 95% CI, 7–98% forA 67-year-old man was admitted with epigastric pain. At the time of the admission, blood pressure was 100/60 mmHg, pulse was 92 beats/min, respiratory rate was 20 breaths/min, and temperature was 37.2°C. The patient was referred to our hospital with general weakness and a weight loss of 10 kg for the last 3 months. Mild epigastric tenderness was revealed during physical examination.3. A comprehensive metabolic blood panel revealed a low albumin level of 2.5 g/dL, low sodium of 130 mEq/L, increased AST/ALT of 131/117 IU/L, and increased alkaline phosphatase of 539 IU/L. Renal functions were normal.His laboratory tests revealed anemia with hemoglobin of 7.6 g/dL and leukocytosis with white blood cell count of 18,380/mmAn electrocardiogram revealed right bundle branch block, with no changes compared to the previous exam. The levels of creatine kinase MB and troponin-I were normal.Abdominal computed tomography (CT) revealed a heterogeneous left adrenal mass 6 cm in diameter, accompanied by hemorrhaging .μg/day ; norepinephrine: 52.1 μg/day ; 17-ketosteroid: 5.76 mg/day ; and metanephrine: 0.47 mg/day .Hormonal testing through 24 h urine collection showed the following results: epinephrine: 4.8 μg/day , were elevated. In addition, plasma ACTH, <1.0 pg/mL , was suppressed, with normal plasma renin activity and aldosterone levels. With the suppression of ACTH and the elevation of urine-free cortisol, we performed a low-dose dexamethasone suppression test to check for Cushing's syndrome. The plasma cortisol level (6.13 μg/dL) showed no suppression at the end of test. We interpreted these results as subclinical Cushing syndrome because he had no symptoms or signs of Cushing's syndrome. 123I-Metaiodobenzylguanidine (123I-MIBG) scintigraphy was performed to check for the presence of pheochromocytoma. For this purpose, 3 mCi of 123I-MIBG was injected and whole body images were obtained after 6 h and after 24 h. From the 24 h images, eccentric uptake by a large left adrenal gland mass was confirmed , 10.42 mg/day , and urine-free cortisol, 491.4  Figures . On the  portion .During preparation of surgery for suspected subclinical Cushing's syndrome and pheochromocytoma, chest X-ray and chest CT  confirmeStarting from 2 weeks prior to the surgery, phenoxybenzamine and propranolol were administered. Laparoscopic left adrenalectomy was performed. The resected tumor measured 8.5 × 7.5 × 5.1 cm; central necrosis and hemorrhaging were present, with involvement of the resection margins . MicroscTwo days after tumor resection, the patient reported dyspnea and developed hypoxia; chest CT confirmed the presence of multifocal pulmonary thromboembolism, bilateral pneumonia, and pulmonary infarction. Mechanical ventilation, intensive care, and administration of antibiotics were continued, but the patient showed pneumonia aggravation, acute kidney injury, and fungemia, with no signs of improvement. The patient's legal guardian refused to continue intensive care and transferred the patient to another hospital for supportive treatment. The patient died 2 months after his initial diagnosis.Pheochromocytoma, along with paraganglioma, is a chromaffin neuroendocrine tumor that secretes catecholamine, which mostly manifests characteristic symptoms including headache, hypertension, and palpitation. However, approximately 10% of patients may show no symptoms, and careful evaluation is necessary, as pheochromocytoma can be life threatening if left undiagnosed . A diagn123I-MIBG scintigraphy is widely used as a functional imaging tool. It has high sensitivity and specificity in diagnosis of neuroendocrine tumors, which, along with anatomical imaging tools such as CT and MRI, is recommended in the diagnosis and localization of pheochromocytoma [123I-MIBG scintigraphy has a sensitivity and positive predictive value of 100%; even in patients with symptomatic pheochromocytoma and normal urinary catecholamine metabolites, positive uptake in 123I-MIBG scintigraphy is seen. Taken together, this demonstrated the effectiveness of 123I-MIBG scintigraphy in diagnosis of pheochromocytoma [Currently, mocytoma . In addimocytoma , 14.123I-MIBG scintigraphy have been reported, even though the mechanism for false positive uptake remains unclear. Several explanations have been proposed in confirmed cases of false positive uptake. For example, neuroendocrine tumors with origins other than a chromaffin neuroendocrine tumor, medullary thyroid carcinoma, and carcinoid tumors can all show uptake of 123I-MIBG, which is believed to occur via intracellular granules through their respective amine uptake mechanisms [123I-MIBG uptake by normal adrenal gland [However, rare cases of false positive uptake in chanisms . Furtheral gland , 7, 16.123I-MIBG into cells from chromaffin neuroendocrine tumors mainly occurs via the norepinephrine transporter and passive diffusion, which is then accumulated in the secretory granules inside cells by vesicular monoamine transporters (VMATs) [123I-MIBG has been reported in large adrenocortical carcinoma, hepatocellular carcinoma, and malignant lymphoma, which is suspected to be related to increased 123I-MIBG uptake by passive diffusion resulting from increased blood flow [123I-MIBG uptake have been reported in mast cell-infiltrated infantile hemangioma, which is believed to be caused by increased expression of VMAT2 by mast cells [The uptake of  (VMATs) . False pood flow . Moreovest cells .123I-MIBG scintigraphy is believed to be the result of increased passive diffusion from increased blood flow and the lesion's location in the adrenal gland. Furthermore, no uptake of 123I-MIBG may have been seen in metastatic lymphadenopathy because, after acquiring a whole body image in the first 6 h, the subsequent topography performed after 24 h focused only on the adrenal gland.In this case, the false positive uptake seen on 123I-MIBG scintigraphy was performed to assess the presence of pheochromocytoma.In the present case, except for weight loss, other symptoms typical of pheochromocytoma such as headache and palpitation were absent; normal levels of catecholamine and metanephrine were found in hormonal testing, with slight elevation seen only in VMA. However, in biochemically silent cases of pheochromocytoma, instead of typical symptoms associated with catecholamine excess, only the symptoms associated with the tumor effect may be present, as seen in the present case . Further123I-MIBG scintigraphy was discovered by chest CT and PET-CT, which led to suspicion of malignant pheochromocytoma. Malignant pheochromocytoma comprises 13~34% of all pheochromocytoma cases, where about one-half shows metastasis at first diagnosis, with the rest undergoing metastasis after a median time of 5.6 years [Metastatic lymphadenopathy that was not seen in .6 years . Althoug.6 years . In the 123I-MIBG scintigraphy in diagnosis of pheochromocytoma is widely known and can be performed in cases where catecholamine excess is not confirmed by biochemical testing, but when clinically suspected. However, despite the fact that 123I-MIBG scintigraphy is an outstanding functional imaging tool, due to the remote possibility of false positive uptake, interpretation of 123I-MIBG scintigraphy and diagnosis of pheochromocytoma should be made with caution. When the degree of 123I-MIBG uptake is low-grade in relationship to a large tumor mass, then a false positive result should be entertained, particularly in the absence of typical clinical and biochemical features of pheochromocytoma.The patient underwent surgical resection for definitive diagnosis and correction of excess hormonal secretion. Unfortunately, two days after surgery, the patient developed pulmonary thromboembolism, pulmonary infarction, and pneumonia, which did not improve. The patient died 2 months after his initial diagnosis. The utility of