H3K27M-mutant diffuse midline glioma is a recently classified unique entity predominantly affecting pediatric patients and rarely adults. The clinicopathologic features in adults remain poorly characterized.A 36-year-old man presented with subacute progressive cognitive and visual deterioration, and hydrocephalus requiring ventricular shunting. MRI revealed a diffusely infiltrating lesion with a gliomatosis cerebri growth pattern, multiple foci of contrast enhancement, and diffuse leptomeningeal involvement. Suboccipital craniotomy with exploration of the posterior fossa revealed a subtle capsular lesion infiltrating into the choroid plexus. Although histologically low-grade, the tumor was found to have an H3K27 M mutation establishing the diagnosis.In spite of diverse clinicopathologic characteristics, H3K27M-mutant diffuse midline gliomas are incurable, WHO grade IV lesions with poor prognosis. We discuss our case in the context of a review of published reports of H3K27-mutant diffuse midline gliomas in adults. Findings late in the disease course may mimic inflammatory or infectious pathologies radiographically, and low-grade infiltrative neoplasms histologically.The diverse clinical, radiographic and molecular features of H3K27M-mutant diffuse midline gliomas in adults remain to be completely characterized. A high index of suspicion is required to avoid missing the diagnosis. Early biopsy and detailed molecular characterization are critical for accurate diagnosis and patient counseling. Many of these cases were previously classified as brainstem gliomas or diffuse intrinsic pontine gliomas (DIPG) . These l2A 36-year-old male presented with two months of nausea, vomiting, headache, bilateral visual loss, hiccups, fatigue, and weight loss. He was found to have communicating hydrocephalus and papilledema requiring ventriculoperitoneal shunting. His visual symptoms recurred one month later, requiring shunt revision. Cerebrospinal fluid (CSF) chemistries and infectious studies were unremarkable. After experiencing one week of improvement, his vision again relapsed.On evaluation, the patient was drowsy, disoriented and blind in both eyes, without other focal deficit. Serum chemistries, blood counts and coagulation parameters were within normal limits. Magnetic resonance imaging (MRI) of the brain and spine was notable for communicating hydrocephalus and transependymal flow of cerebrospinal fluid A. There The differential diagnosis at this point included infectious etiologies such as meningitis and encephalitis; inflammatory conditions such as neurosarcoidosis and primary angiitis; and neoplastic etiologies such as diffuse astrocytoma with gliomatosis cerebri growth pattern and leptomeningeal spread. Contrast-enhanced computed tomography (CT) scans of the chest, abdomen and pelvis were unremarkable. Infectious and neoplastic assays on CSF were negative across multiple samplings. Another shunt revision was performed, with concurrent biopsy of the sural nerve and muscle. Biopsy results were negative, and the patient was thus offered craniotomy with exploration of the posterior fossa and open surgical biopsy.A standard midline suboccipital craniotomy was performed. Upon opening the posterior fossa dura, thickened arachnoid was encountered and carefully divided. The cerebellar tonsils were split in order to expose the fourth ventricle. A small capsule in the wall of the fourth ventricle was biopsied; no other grossly abnormal area was encountered.Histopathologic analysis of the fourth ventricular lesion showed neoplastic spindle cell proliferation with spread into the choroid plexus. Cells were positive for S-100 and GFAP, and rarely for CD68. Molecular genetic analysis showed an H3K27 M missense mutation in the H3F3A gene and a truncating mutation in the NF1 gene. Chromosomal analysis revealed multiple copy number losses , one copy number gain (1p/q), monosomy 13, and one large region of loss of heterozygosity at 3q. An integrated diagnosis of H3K27M-mutant diffuse midline glioma was thus established.Postoperatively the patient additional episodes of shunt failure requiring revisions. Due to continued clinical deterioration, his family elected to pursue palliative measures including whole brain radiation. He later suffered multiple episodes of aspiration pneumonia, and expired due to septic shock 8 months after initial symptom onset.3The 2016 WHO classification of central nervous system tumors incorporated molecular parameters into the classification of diffuse gliomas, a dramatic change from the nearly century-old classification based exclusively upon histological features . In the Published reports of adult H3K27-mutant diffuse midline gliomas are summarized in Reported radiographic appearances were also highly heterogeneous, though infrequently described in detail. Greater than 50% of cases displayed contrast enhancement on imaging. Leptomeningeal enhancement similar to our report was described in only two cases ,21,23. MH3K27M-mutant gliomas display a wide spectrum of histologic phenotypes, and are classified as WHO grade IV irrespective of histologic grade ,21,28. TUnder the WHO classification, H3K27M-mutant gliomas are by definition IDH-wildtype and lack 1p/19q co-deletion. These gliomas are instead characterized by a distinct mutation in histone H3 genes . MutatioThe majority of cases are managed with surgery, radiation and alkylating chemotherapy (primarily Temozolomide). The role of surgery is primarily for diagnosis, owing to the surgically inaccessible location of most lesions. Prognosis remains dismal even with timely and optimal treatment; overall survival is typically 7–11 months in children ,33 and 84H3K27M-mutant diffuse midline gliomas predominantly affect pediatric patients, but may rarely present in adults. Our comprehensive literature review of adult cases highlights the need for further studies to characterize the diverse clinical, radiographic and molecular features of these lesions in adults. These gliomas must remain in the differential diagnosis of any new brain lesion affecting midline structures. Presentation late in the disease course may mimic gliomatosis cerebri growth pattern radiographically, and benign infiltrative neoplasms histologically. Early surgical biopsy with attention to subtle anatomic abnormalities, intraoperative histopathologic analysis, and molecular genetic analysis including histone mutation screening are critical for accurate diagnosis, management and patient counseling.This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.This is a case report; therefore, it did not require ethical approval from ethics committee.Exhaustive attempts were made to contact the family of the deceased patient. The paper has been sufficiently anonymized not to cause harm to the patient or the family. We have uploaded a separate document attesting to this, signed by the head of our medical team.All authors: writing the paper, design and data collection, data analysis and interpretation.Not applicable.Hoi Sang U, MD; and Mihir Gupta, MD.Not commissioned, externally peer-reviewed.The authors report no conflicts of interest.


Traumatic diaphragmatic rupture is uncommon, life threatening and remains a diagnostic and radiographic challenge. Diagnosis is frequently delayed, which may result in a late intervention with a potential catastrophic outcome. We report a case of an acute diaphragmatic laceration in a 40-year-old woman, with a personal history of bipolar disease, admitted on the emergency department after falling from a nine-meter building. During initial evaluation, the plain chest radiograph showed multiple rib fractures associated with a significant left pneumothorax. It also showed an elevated left diaphragm with a suspicious gastric shadow in the left hemithorax. Computed tomography confirmed the diagnosis of a left-sided diaphragmatic laceration and the patient was advised surgical intervention. During laparoscopy, a 7 cm rupture of the left hemi-diaphragm with herniation of the stomach was identified. The hernia was reduced laparoscopically and the defect repaired with interrupted, non-absorbable, sutures. The patient had an uneventful recovery and remained well at a 3-month follow-up visit. Emergency repair of the diaphragm is usually performed via a thoracotomy or/and laparotomy. However, if the patient is hemodynamically stable and major organ injuries have been excluded, a laparoscopic approach can be considered safe and effective. Diaphragmatic rupture is an infrequent and life-threatening complication of trauma. As the signs and symptoms of acute diaphragmatic trauma are often masked by severe concomitant injuries to other organs, a high índex of suspicion is warranted for clinical diagnosis, especially in the absence of pathognomonic radiographic findings ,2. DiagnA 40 year-old woman was admitted to the emergency department after falling from a nine-meter building. The patient complained of left hemithorax pain and shortness of breath, left leg and lower back pain.On initial evaluation, her vital signs were stable. Chest evaluation revealed respiratory distress on 6L/min oxygen support, pain on palpation and loss of breath sounds on the left hemithorax. The abdomen was mildly tender to deep palpation and she could not move her left leg due to pain. Aside from having a bipolar disorder, followed by regular psychiatry appointments, she lacked a significant past medical history.The patient was evaluated and stabilized in the emergency room. Chest radiograph showed multiple rib fractures and a left pneumothorax. It also showed an elevated left diaphragm with suspicious gas pattern of the stomach Figure .The chest radiograph was compared to past x-rays taken during a previous hospital admission and none of the described alterations were present before.Computed tomography (CT) scan of the thorax and abdomen revealed the presence of a left pneumothorax with associated costal fractures. It also showed an acute rupture of the left hemi-diaphragm causing herniation of the stomach and subsequent lung atelectasis Figure , 3.No other thoracic or abdominal injuries were reported. Other minor associated injuries such as left tibia fracture were identified. The patient was advised for a minimally invasive surgical intervention and taken to the operating room.After placing a left chest tube in the second intercostal space, an exploratory laparoscopy was performed. The greater omentum and the stomach were found within the left hemithorax, protruding through a seven-centimeter diaphragmatic defect .After reducing the hernia contents, a tension-free repair was performed with interrupted silk suture; the use of an absorbable mesh was considered unnecessary. No other signs of injury were identified upon final inspection of the abdominal cavity. The patient’s postoperative course was uneventful with complete expansion of the right lung Figure .The patient was discharged on the fifth postoperative day after psychiatric and orthopedic evaluation. On the third month follow-up, the patient was clinically asymptomatic and no evidence of recurrence was identified on the x-ray.Diaphragmatic hernia is a herniation of the abdominal organs or tissues into the thoracic cavity. Traumatic diaphragmatic injuries are rare and usually occur after thoracic or abdominal blunt (2.9%) or penetrating 3.4%) trauma % trauma ,6.However, the precise incidence of this injury is likely to be under-reported. Diaphragmatic injuries, in the absence of acute diaphragmatic hernias, are often missed by diagnostic imaging . The diaThe possibility of a diaphragmatic injury should always be considered in the context of rapid deceleration or crush injuries . There aMost of the traumatic diaphragmatic injuries (80-90%) occur in the left diaphragm because the left diaphragm is congenitally weaker than the right diaphragm which is protected by the liver . The cliThe symptoms and signs are strongly associated with the size of the diaphragmatic defect, herniated organs and the existence of pulmonary disease . During However, a chest radiograph may not be useful in some cases as signs are often masked by associated lung contusion, hemothorax, pneumothorax, pleural effusion, atelectasis, emphysema and non-specific elevation of diaphragm . BetweenCT scan of the chest has become an essential tool for the evaluation of a hemodynamically stable trauma patient. In the absence of an acute hernia, CT scans offer little benefit compared with conventional plain radiographs, as the sensitivity of the CT for the diagnosis of isolated diaphragmatic injury is limited. However, in the presence of herniation of abdominal organs into the thoracic cavity, the sensitivity of oral contrast-enhanced CT scan is close to 95%. It is especially helpful if the plain chest radiograph is obscured by the presence of a hemothorax or a lung contusion ,7.Traumatic rupture of the diaphragm is considered an indication for surgical repair, especially in symptomatic patients. The onset of complications carries highest mortality and morbidity rates. This makes emergency surgery mandatory .Two principles must be followed when repairing acute traumatic diaphragmatic hernias: complete reduction of the herniated organs back into the abdomen and watertight closure of the diaphragm to avoid recurrence. Repair with non-absorbable simple sutures is adequate in most cases and the use of a mesh should be reserved for chronic and large defects. In some series, suture with absorbable sutures are associated with a higher rate of recurrence .In patients with giant diaphragmatic hernias, synthetic or biologic grafts can be used for a tension free repair and prevention of recurrence . It shouLaparotomy or thoracotomy are the traditional surgical approaches for patients with diaphragmatic rupture, the choice being largely dependent on the skill set of the surgeon involved ,4.However, given the high rate of associated injuries to intra-abdominal organs, it is generally recommended to approach the diaphragmatic injury through an abdominal approach rather than thoracic . LaparosFor these reasons, laparoscopic approaches for repair of hernias have gained in popularity being an excellent diagnostic and therapeutic approach in the setting of suspected diaphragmatic rupture ,5. OutcoDiaphragmatic injury is a rare entity and carries a significant morbidity and mortality, usually from associated injuries and delayed diagnosis. There are no specific signs and symptoms for diagnosing diaphragmatic rupture, the key being a high index of suspicion.Reports of laparoscopic repair of traumatic diaphragmatic rupture are uncommon. However, in hemodynamically stable patients, laparoscopy should be considered as a diagnostic and therapeutic approach. It is safe and effective in repairing the diaphragm of selected patients following blunt abdominal trauma.


Primary spinal melanoma (PSM) is a rare primary central nervous system melanoma with limited literature. A 30-year-old male presented with one year of progressive bilateral leg weakness and back pain. Physical examination revealed slightly decreased power and deep tendon reflexes of the lower extremities, decreased sensation at the level of T10, and normal anal sphincter and plantar reflexes. Magnetic resonance imaging (MRI) scan of the thoracolumbar spine revealed a hypointense lesion on T2-weighted and a hyperintense lesion on T1-weighted imaging at the level of T10 with mild extensions. The lesion was causing a mass effect on the spinal cord. The patient underwent laminectomy and near-total excision which showed a black, firm-to-hard, scarcely vascular extradural tumor extending from T10 to T11 that adhered to nerve roots. Histopathological examination and immunostaining with S-100 and Melan-A stains confirmed the diagnosis of malignant melanoma. Other imaging studies like brain computed tomography (CT) and positron emission tomography/computed tomography (PET/CT) scans, and chest X-ray were normal. On follow-up, the patient reported improvement in the power of his lower limbs with intact sensory function and sphincters. The first radiotherapy session was scheduled for six weeks postoperatively. There was no recurrence at a two-year follow-up. The possibility of a melanocytic tumor should be considered for a spinal lesion with paramagnetic properties as early surgical intervention is important for diagnosis and improved survival. Malignant melanoma is a cancer of melanocytes that can arise from any melanin-containing cell. Primary central nervous system (CNS) melanoma, however, is rare. It comprises only 1% of melanomas which include blue nevus, meningeal melanocytoma, melanotic schwannoma, and malignant melanoma , 3.Due to the rarity of PSM, the literature on the epidemiology, natural history, diagnostic/therapeutic approach, and prognosis is not vast. To our knowledge, we are reporting the first case of extradural PSM with intradural extension from Pakistan.A 30-year-old male patient presented with a complaint of bilateral leg weakness for one year. The weakness started gradually in the right leg followed by the involvement of the left leg. It was associated with progressive middle back pain that was aching in nature, 4/10 in intensity, and worse at night. He denied any stiffness, numbness, tingling, urinary/bowel incontinence, skin changes, or fever/chills. Personal, family, and social histories were non-contributory.Physical examination of bilateral lower and upper extremities revealed power grades of 3/5 and 5/5, respectively. The patient had slightly diminished deep tendon reflexes (DTRs) in the ankles and knees bilaterally. Neurological examination of the back revealed diminished sensations at the level of T10. Plantar and anal sphincter reflexes were intact. Skin and eye examinations were normal. Due to chronic leg weakness and nocturnal back pain, the patient was admitted to the hospital for imaging studies.DiagnosisMagnetic resonance imaging (MRI) scan of the thoracolumbar spine revealed a 1.5 x 1.2 cm hypointense lesion on T2-weighted (T2-W) imaging and a hyperintense lesion on T1-weighted (T1-W) imaging at the level of T10-T11 with mild intradural and extradural extensions  is the best imaging modality to establish a preliminary diagnosis of spinal tumors. Malignant CNS melanoma characteristically appears hyperintense on T1-W and hypointense on T2-W MRI. In contrast studies, mild homogenous enhancement is seen . WoodrufThere is no standardized treatment for PSM, but the therapeutic approach differs greatly from that of metastatic melanoma. Surgical resection of the tumor is the treatment of choice. The goal of surgery is therapeutic as well as diagnostic. Adjuvant radiotherapy may help in limiting tumor dissemination . It is nDue to limited data, the prognostic parameters of PSM and its overall survival are not certain. A retrospective study reported that tumor dissemination and chemo/radiotherapy without surgical resection were associated with the worst prognosis in PSM cases . In patiIn literature, PSM has been reported as an indolent tumor as compared to metastatic melanoma. The average survival in PSM patients after surgical resection was six years and seven months . HoweverWe must be cautious with the diagnosis of PSM. The possibility of melanocytic tumor should be considered for a spinal lesion with paramagnetic properties. Our case emphasizes that early surgical intervention to obtain a histological diagnosis is important and improves survival in patients with PSM.


Idiopathic chronic ulcerative enteritis (ICUE) is a very rare disease with high mortality. Because of clinical rarity, several small case reports have been published and there is a lack of large sample study. Preoperative definite diagnosis is difficult. Although definite treatment for ICUE is radical surgical resection, surgical decision in operative field is difficult.A 77-year-old man came to the emergency department with complaints of a 1-day history of abdominal pain and abdominal distension. Abdominal computed tomography revealed ileus and focal free air. Laparotomy revealed multiple small bowel tiny perforations in the ileum. The serosa surface in the whole small bowel had small multiple yellowish tiny discolored lesions. Despite the presence of multiple mucosal ulcers in entire small bowel, the ileum including perforation site was resected segmentally. Microscopically, mucosal ulcers in resected small bowel demonstrated transmural inflammation, no granuloma, and no lymphoid aggregates. These features were consistent with a diagnosis of ICUE with panenteritis and perforation. After surgery, the patient’s general condition gradually aggravated. Unfortunately, the patient died of multiple organ failure on post-operative day 14.Surgically, the decision including resection range, anastomosis or enterotomy becomes difficult in ICUE with panenteritis. According to recent 40 year’s revised data, the post-operative mortality of ICUE is about 53.4%. Although ICUE is rare, its recognition is important for appropriate diagnosis and treatment. Retrospective multicenter case studies are required to determine proper treatment and improve prognosis. Idiopathic chronic ulcerative enteritis (ICUE) is a rare disease and diagnosis of ICUE is very difficult before laparotomy. The etiology of ICUE remains unknown.Historically, multiple diffuse nonspecific nongranulomatous small intestinal ulcer without underlying disease was first described in 1949 as a kind of ulcerous jejuno-ileitis [Additionally, ICUE is defined in terms of several different descriptive phrases including ulcerative jejunitis, ulcerative jejunoileitis, nongranulomatous ulcerative jejunoileitis, ulcerative enteritis, and nonspecific ulcerative duodenojejunoileitis , 3.ICUE patients have chronic gastrointestinal symptoms. However, most of the ICUE cases remained undiagnosed until laparotomy. Generally, laparotomy is performed in emergency settings because of intestinal obstruction, perforation, and bleeding. Finally, after laparotomy, the diagnosis of ICUE becomes feasible in most of the cases of ICUE.In the current study, we present a case of a 77-year-old male patient diagnosed with ICUE with panenteritis and perforation. Despite surgical treatment, post-operative mortality occured.Also, we have included a review of the literature.This study was approved by the Institutional Review Board of Yeungnam University Medical Center (IRB No. 2020–01-037). The patient provided written informed consent regarding the publication of case details at admission.A 77-year-old man came to the emergency department with complaints of a 1-day history of abdominal pain and abdominal distension.His family history about gastrointestinal disorder was non-specific. His past-medical history including non-steroidal anti-inflammatory drug (NSAID) medication was non-specific.The initial blood pressure was 160/70 mmHg, heart rate was 86/min, respiratory rate was 20/min, and body temperature was 36.9 °C at admission. Initial laboratory findings revealed slight elevation of leucocytes and hemoglobin at 11.9 g/dL.The bowel sounds were silent. Physical examination revealed tenderness in the periumbilical area with rebound tenderness and guarding.Abdominal computed tomography revealed ileus with edematous small bowel loops, mild ascites, non-specific mass lesions, and focal free air in the right upper quadrant Fig. .Fig. 1AThe results led us to consider peritonitis due to small bowel perforation. The surgical procedure was indicated and emergency exploration was performed thereafter.Laparotomy revealed multiple small bowel tiny perforations in the ileum. The perforation sites were in the form of very small holes and measured about 0.1 ~ 0.2 cm in size. Interestingly, all the perforation sites were located in the mesenteric border in the small bowel  is defined as multiple diffuse nonspecific nongranulomatous small intestinal ulcerative disease with unknown etiology.Most of the ICUE patients in previously reported literature had chronic gastrointestinal symptoms.In the early stage of ICUE, the symptom is ambiguous. However, general presentations in ICUE are weight loss, chronic abdominal pain, diarrhea, melena, hematochezia, a positive fecal occult blood test, malnutrition, iron deficiency anemia, hypoalbuminemia, and so on , 5.However, clinical features vary including the asymptomatic case, chronic gastro-intestinal symptoms, and acute abdominal pain. Although these symptoms are helpful for suspicion of small bowel disease, they are of no aid for early diagnosis of ICUE. In some cases without a history of chronic symptoms, diagnosis of ICUE has been made , 6.Under conditions of the above-mentioned presentations, several diagnostic examinations are recommended.About 40 years back, per-oral jejunal biopsy and small bowel series were performed for the diagnosis of small bowel abnormality .However, with recent developments of radiologic tests and endoscopic equipment, gastro-endoscopy, colonoscopy, double-balloon enteroscopy, computed tomography, and capsule endoscopy are considered for the diagnosis , 7. In KHowever, these radiologic and endoscopic examinations are not effective in the definite diagnosis of ICUE. Most of the ICUE cases remain undiagnosed until laparotomy.Finally, the definite diagnosis of ICUE is made by pathological evaluation of resected small bowel specimens after laparotomy.When a patient has small bowel obstruction, perforation, bleeding, chronic malnutrition or weight loss related gastrointestinal function, laparotomy is performed , 6, 8–10Among 43 patients, bowel perforation developed in 14 patients and bowel obstruction developed in 19 patients , and chronic nonspecific multiple ulcers (CNSU).Behcet’s disease is characterized by the presence of ileocecal ulcer along with transmural inflammation and crater-shaped ulcer margins. Also, Behcet’s disease has a triad of symptoms consisting of aphthous stomatitis, genital ulcers, and ocular symptoms . HoweverNSAID-induced enteropathy has a characteristic narrow ileal stenosis that is called as ‘diaphragm disease’ . HoweverCMUSE is characterized by the presence of small intestinal stricture and superficial mucosal or submucosal ulcer in the small bowel , 12. HowChronic nonspecific multiple ulcers (CNSU) are different from ICUE. CNSU is limited to only mucosal and submucosal small intestinal ulcers. However, ICUE has variable ulcer depth ranging from mucosa to transmural depth including perforation .Medical treatment including steroid or gluten-free diet is not effective in ICUE , 6.Definite treatment for ICUE is radical surgical resection. Furthermore, repeated laparotomy is required frequently because of symptom recurrence .Despite surgical treatment, post-operative aggravation of the ulcer may develop like intestinal bleeding or perforation. These aggravations may preclude reoperation and lead to death , 4.For analysis of prognosis in ICUE, we reviewed the clinical data of the previously reported cases of ICUE and our case Table .The frequently cited post-operative mortality rate of ICUE was 62.5% in the report published 40 years back [In conclusion, we present a case of ICUE with panenteritis and perforation treated by wide small bowel resection. The diagnosis of ICUE with panenteritis is very difficult before laparotomy. Surgically, the decision including resection range, anastomosis or enterotomy becomes difficult in ICUE.Despite definite surgical treatment, postoperative intestinal bleeding developed and the patient died of multiple organ failure. According to recent 40 year’s revised data, the post-operative mortality of ICUE is about 53.4%. Although ICUE is rare, its recognition is important for appropriate diagnosis and treatment. Retrospective multicenter case studies are required to determine proper treatment and improve prognosis.


Mesenteric cysts were first described in 1507 and since then remain as a rare intraabdominal pathology. The etiology of this kind of tumor is still unclear and the classification remains controversial. They are usually asymptomatic, but can also cause acute abdominal pain and sometimes need emergency surgical approach. Clinical history, physical exam and complementary tests do not always provide diagnosis, which in many cases is made after surgery. Surgical management with complete excision of the cyst is the gold standard treatment. Laparoscopy technique should be preferred whenever is possible. The knowledge of these rare tumors is important for considering the correct approach. The goals of this article is to describe a case report of mesenteric cyst presented with acute abdominal pain at the emergency and do a brief literature review about this entity. •Mesenteric cysts are a rare cause of intraabdominal mass.•Mesenteric cyst should always be considered it whenever the patient has an intraabdominal palpable mass.•Computed tomography of the abdomen can help find the diagnosis in many cases.•Even when mesenteric cyst is diagnosed incidentally, surgery should always be indicated in order to prevent complications.•Surgery is the treatment of choice and recurrence rate is low when the correct treatment is performed. It was 2A 33-year-old male patient with no significant past medical history presented to the Emergency Department due to severe abdominal pain during the last five days. The pain was located in the left flank, radiated to the back, associated with difficult evacuation and one episode of bloody vomiting. He denied weight loss, fever and other constitutional symptoms.On physical examination a large abdominal mass could be felt occupying from the epigastrium to the hypogastrium, in addition to both flanks, being slightly lateralized to the left, with well-defined margins, fibroelastic consistency, besides being mobile and slightly tense and painful on palpation, probably due to its great extension. There were no signs of peritonitis.Blood tests including full blood count serum electrolytes, canalicular enzymes, bilirubin, pancreatic enzymes and liver and kidney function tests were performed which, besides a hemoglobin level of 10.5g/dL (reference range: 14–18 g/dL), were unremarkable and showed no other abnormalities. Prothrombin time, partial thromboplastine time and INR  were slightly increased. As for tumor markers, Carcinoembryonic Antigen (CEA) level was 0,52ng/mL  and Carbohydrate Antigen 19-9 (CA 19-9) was 3,92U/mL (reference range: <35U/mL).During the investigation, a computed tomography (CT) was requested, which exhibited a large predominant cystic lesion, with solid component in posterior region, measuring 17, 8 × 16 × 10, 9cm, located mainly in the mesogastrium causing compression of intestinal loops, suggesting the diagnosis of mesenteric cyst .Fig. 1AbThe resection of the cystic lesion was indicated. Laparoscopic approach was not attempted due to the size of the lesion. At surgical approach through median incision, a large cyst with peritoneum and small bowel adhesions was found. After anterior and lateral dissection and complete liberation of the colon, the anterior wall of the cyst was incised and 1.500 mL of a dark brown fluid was drained out and sent for further analysis. The posterior wall of the cyst was firmly connected to the root of the mesentery, adjacent to the middle colic vessels, making it impossible to safely resect it and was opted to perform cystic marsupialization .Fig. 2InThe total operative time was 125 minutes and the patient was discharged home three days after the procedure without intercurrences.In the analysis of the cyst fluid, hypocellular cytological preparations exhibited rare lymphocytes and histiocytes, on a sero-hemorrhagic background. No epithelial elements or cytological signs of malignancy were identified in the material examined. Histopathological examination of cyst capsule revealed a dense fibrous connective wall with indistinct epithelial lining, besides of frequent thin-walled congested vessels, lymphocytic inflammatory process with associated hemosiderophages and no signs of malignancy .Fig. 3CyThe findings were consistent with the diagnosis of benign cystic mesothelioma of the mesentery. At a post-operative follow up period 3 months, there were no significant symptoms and no clinical evidence of recurrence.3Although the first description of mesenteric cyst was in the 16th century ,9, its oMesenteric cysts can be located anywhere in the mesentery from the duodenum to the rectum ,7,9, andThe incidence of malignant cysts is low (less than 3% of cases) ,9,13 andMany authors consider mesenteric, omental, and retroperitoneal cysts as one group because they derive from the same embryological structures, while others believe that the retroperitoneal lesions represent another group of pathology .Several classifications have been reported. The most widely recognized divides mesenteric cysts into four groups based on clinical and etiological features: 1) embryonic and developmental; (2) traumatic; (3) neoplastic, and (4) infectious  embryoni.They can also be differentiated into lymphatic cysts and lymphangiomas or simple mesothelial cysts and benign cysts. Simple lymphatic and mesothelial cysts usually remain stable and asymptomatic over time, as lymphangiomas and benign cystic mesotheliomas may have aggressive and invasive properties. Histopathologically, flat endothelial cells characteristically line cysts of lymphatic origin with a wall containing smooth muscle fibers, lymphoid tissue, and/or occasional foam cells. In contrast, flat, cuboidal, or columnar cells line cysts of mesothelial origin and their wall is fibrous without any lymphatic structures. The distinction between simple lymphatic cysts and lymphangiomas or between simple mesothelial cysts and benign cystic mesotheliomas is based on macroscopic characteristics. Simple cysts are usually small (1–5 cm) and unilocular, whereas lymphangiomas and benign cystic mesotheliomas are large and include multiple cysts of several sizes. Occasionally, their gross appearance may be similar and the distinction is not possible .Mesenteric cysts can be single or multiple and uni or multi-loculated and the contents of the cyst vary from clear to milky or dark brown depending on the location of the cyst and the presence or absence of hemorrhage .Although often asymptomatic, 10% of the patients present as an acute abdomen . They caThe size of the cysts and age of the patient influence the clinical presentation ,9. AbdomChildren usually present with acute abdomen, while adults have more indolent symptoms . The cysThe clinical history and physical exam may suggest the diagnosis, but in many cases the diagnosis is made by the exclusion of other causes or only after surgery . The diaLaboratory tests contribute little to the diagnosis . UltrasoThe surgical treatment is the complete excision of the mesenteric tumor ,13 eithePartial excision is associated with recurrence and morbidity ,5,7,9,114Although mesenteric cysts are a rare condition, it is important the knowledge of these tumors, especially in cases where an intraabdominal mass is felt on the physical examination, so the properly treatment can be performed. If physical examination and imaging tests fail to provide the correct diagnosis, surgery should be indicated for the purpose of diagnosis and treatment, with laparoscopy being the preferred technique whenever possible.Not commissioned, externally peer-reviewed.Since we didn't use study of others medical records besides the patient of the case, it was not necessary to submit the paper to the Ethics Committee of our institution. However, the patient consented to carry out this work and all ethical standards have been respected in this work.The authors declare that did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.The patient consented to carry out this work and all ethical standards have been respected in this work .Nubyhelia Maria Negreiro de Carvalho - nubyhelia@gmail.com.The authors declare that they have no competing interests and this research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.


Basidiobolus ranarum. It frequently causes cutaneous infection, but it rarely infects visceral tissues in humans. Here, a 39-year-old previously healthy woman presented with severe left-sided abdominal pain and weight loss. She had visited several hospitals and had provisionally been diagnosed as having either a retroperitoneal malignancy or retroperitoneal fibrosis before being referred to our hospital. Abdominal computerized tomography and biopsy of the retroperitoneal mass revealed retroperitoneal basidiobolomycosis infection. She was started on antifungal treatment. This led to significant improvement, without surgical intervention. Gastrointestinal basidiobolomycosis can present in many forms, commonly involving the colon and liver with multifocal inflammatory masses. Nonetheless, retroperitoneal basidiobolomycosis presentation is extremely rare and should be considered in the differential diagnosis of a retroperitoneal mass with eosinophilia.Basidiobolomycosis is an uncommon emerging fungal infection caused by  Basidiobolus ranarum [Basidiobolus ranarum are possible routes of transmission of gastrointestinal basidiobolomycosis (GIB) [Basidiobolomycosis is a rare emerging fungal infection, which was first described in humans in 1956, and is caused by  ranarum . This or ranarum . The mod ranarum ,5. IngesGIB is uncommon and affects young and immunocompetent individuals, particularly children . It typiDiagnosis of GIB is difficult to make in the initial presentation due to the non-specific nature of the abdominal symptoms and radiological findings, which usually mimic malignancy, inflammatory bowel disease, or diverticulitis. The key laboratory finding is eosinophilia and the histopathological appearance of fungal hyphae surrounded by eosinophilic infiltration (Splendore–Hoeppli phenomenon) in an individual from an endemic areas ,10. HereA previously healthy 39-year-old woman from the southern part of Saudi Arabia, who had no medical conditions of note other than hypertension, for which she was being treated with amlodipine, presented to our hospital with progressive, severe, left-abdominal pain and a history of weight loss over the previous two months without history of fever. She had had a laparoscopic removal of an ovarian cyst two years prior to her presentation. She had no history of trauma or of any unusual habits. She had sought care from several other hospitals before being referred to our hospital for further management as a case of retroperitoneal fibrosis. The physical examination showed no abnormalities and no palpable masses.9 cells/L, eosinophils 15.7%, absolute eosinophils 0.86 × 109 cells/L, erythrocyte sedimentation rate (ESR) 101 mm/h, and C-reactive protein (CRP) 8.63 mg/L. Additionally, immunological testing revealed that her antinuclear antibody (ANA) was negative, and her immunoglobulin G (IgG) subclasses were elevated as follows: IgG1 12.5, IgG2 9.49, IgG3 1.76, and IgG4 6.7 g/L.We initially worked-up the patient as a case of retroperitoneal fibrosis. The initial laboratory results revealed white blood cells (WBC) 5.47 × 10A computerized tomography (CT) scan of her abdomen  revealed9 cells/L (compared to 0.86 × 109 cells/L), ESR 42 mm/h (compared to 101 mm/h), and CRP 0.45 mg/L (compared to 8.63 mg/L). Currently, she is still on treatment and undergoing follow-up.We performed CT-guided biopsy of the mass. The histopathological evaluation revealed broad and branching fungal hyphae with presence of eosinophilic sheets, an appearance characteristic of basidiobolomycosis . We initGIB is an emerging fungal infection of visceral and gastrointestinal tissues. The first case of GIB was reported in a Nigerian child in 1964 . Most ofThe diagnosis is difficult because of its non-specific clinical presentation, which is usually an insidious onset abdominal pain, fever, weight loss, diarrhea, or the presence of an abdominal mass. The most commonly affected abdominal organs are colon (84%), small bowel (32%), liver (21%), and rarely, other organs such as the stomach may be involved ,12,15. MPeripheral blood eosinophilia was present in most reported cases. Culture is required for the definitive diagnosis, the majority of cases are diagnosed based on the characteristic histopathological appearance of the infected tissue, which shows fungal hyphae surrounded by eosinophilia (Splendore–Hoeppli phenomenon) . RecentlThe radiological manifestations of basidiobolomycosis vary according to the organ affected. CT is the most common radiological modality used for diagnosis. The most common abdominal finding is colonic masses. Masses in the liver and loops of small bowel loops are also commonly seen. Less frequent manifestations include involvement of the kidneys, gallbladder, pancreas, and retroperitoneum . AdditioThe prevalence of GIB in the southwestern region of Saudi Arabia (Tohama) is increasing, primarily due to an increased awareness of the disease. Therefore, the diagnosis should be considered in any patient from this endemic area who present with an inflammatory abdominal mass. GIB has a case fatality rate of up to 20% according to an analysis of retrospective data . Hence, The treatment of choice for abdominal basidiobolomycosis is still a matter of debate. A combination of surgical resection of the affected tissue followed by long-term antifungal medication has been the preferred treatment previously . HoweverGIB can present in many forms, commonly involving the colon and liver with multifocal inflammatory masses. Nonetheless, retroperitoneal basidiobolomycosis presentation is extremely rare and should be considered in the differential diagnosis of a retroperitoneal mass with eosinophilia.


Gliosarcoma is an unusual subtype of glioblastoma multiforme. Its characteristic features are biphasic configuration, constituting a definite, separate glial and sarcomatous differentiation, on histological evaluation. Herein, we present a rare case of Gliosarcoma that had presented only once in our center in last 13 years. A 60 years old, diabetic, hypertensive male patient came to e emergency department with disturbed level of consciousness and right sided hemiplegia which was progressive over four days. On examination he was, conscious, unoriented in time, person or place, his mouth deviated to left and vitally stable. After initial evaluation, CT scan and MRI were advised. These showed a complex left parieto-occipital heterogeneous mass lesion with cystic and solid components, measuring approximately 5.2x4cm. The mass lesion was seen displacing the occipital horn anteriorly and inferiorly with probable extension into the lateral ventricular cavity. There was no associated midline shift or definite herniation. The lesion was diagnosed as highly suggestive of brain tumor with a differential diagnosis of glioblastoma multiforme or ependymoma. Blood picture revealed a rapidly increasing level of anemia. Surgical intervention comprising left parieto-occipital craniotomy and near total resection of the tumor was carried out. On histopathological and immunohistochemical evaluation the diagnosis of GS was established. A plan of a combination of adjuvant chemotherapy and radiation was formulated that was however, declined by the family. On regular follow up, the patients clinical state rapidly deteriorated with persistence of seizures and requirement of repeated blood transfusions. The patient finally passed away after eighth months. It is b in 1955 . Initial in 1955 -6. In 20 in 1955 . It is l in 1955 . GS has  in 1955 -9.60 years old male, known case of hypertension and diabetes was brought to the emergency department with disturbed level of consciousness and right sided hemiplegia which was progressive over four days. There were no witness’s seizures. On examination, he was afebrile, conscious, not oriented to time, person or place and his mouth deviated to left side but vitally stable. The blood picture was normal besides hemoglobin level which was initially 11g/dl (13-17g/dl) but rapidly came to a low level of 8 g/dl. The power and sensation could not be assessed due to aggressive behavior of the patient. On plain CT scan of the head , there wet al. [Gliosarcoma is an unusual morphological sub type of glioblastoma, with slight predominance in males, age ranging from the sixth to seventh decades and a predilection for the temporal lobes, although it can also affect the frontal, parietal and occipital lobes ,5,8-10 Set al. . The patGliosarcoma, a histological biphasic tumor showing separate glial and sarcomatous components, is a very rare clinicopathological entity that was diagnosed only once in the last 13 years in our center. It is associated with a very aggressive clinical course with patient’s rapid deterioration and poor outcome. The patients mean survival rate averages less than a year even with a combination therapy comprising radio and chemotherapy.


Giant adrenal myelolipoma is a rare, benign, sizable, mesenchymal tumor. Preoperative differential diagnosis from retroperitoneal liposarcoma may be challenging. A 66-year-old female patient was admitted because of a sizable tumor at the right retroperitoneal space, incidentally discovered during abdominal ultrasonography for screening purpose. Preoperative imaging studies were indicative for the diagnosis of a giant adrenal myelolipoma (11.7 × 12.9 cm in size); however, a retroperitoneal liposarcoma could not be excluded. We decided to proceed with tumor’s surgical removal by using laparoscopic transperitoneal approach and three-dimensional high-definition camera. Intraoperatively, the tumor did not infiltrate surrounding tissues and was surrounded by a thin capsule under which there were sparse, orange-colored spots that resembled adrenal cortex. This finding reinforced the initial and most possible diagnosis of adrenal myelolipoma and we easily enucleated the mass. Postoperative course was uneventful, and the patient demonstrated no recurrence on imaging six months postoperatively. Histology confirmed the diagnosis of giant adrenal myelolipoma, measuring 16.5 x 15 x 6.5 cm. Adrenal myelolipomas (AMLs) are rare, benign, non-secretory, mesenchymal tumors, composed of mature adipose and myeloid tissue. Size of the tumor is usually less than 4 cm, but it can outreach 10 cm and in this case the tumor is characterized as giant. The diagnosis of AML is indicated by clinical presentation, although it is usually asymptomatic, and by imaging studies and is confirmed by pathological examination. The treatment of choice of giant AML is open surgical removal, but there are numerous reports of giant AMLs which were successfully removed by using minimal invasive approaches  during the last years [The aim of our study is to present the macroscopic appearance of a giant AML as it was recognized intraoperatively during the laparoscopic excision and to underline the feasibility of successful removal of this kind of tumors by using minimal invasive approaches.In February 2019, a 66-year-old female patient was admitted to the Department of Urology of Korgialenio-Benakio Hellenic Red Cross Hospital, Athens, Greece, because of a sizable tumor at the right retroperitoneal space, incidentally discovered during abdominal ultrasonography (U/S) for screening purpose two weeks ago. Her medical history included hypertension, dyslipidemia and hypothyroidism. The patient was asymptomatic, clinical examination was unremarkable, while blood chemistry and adrenal hormones were in the normal range.Preoperative U/S described the presence of a heterogeneous, solid, 11 cm in size mass below liver’s right lobe. Preoperative abdominal computed tomography (CT) scan demonstrated a 12.9 × 11.7 cm in size, encapsulated, hypodense, solid, right retroperitoneal mass, containing mainly fat as well as interspersed soft-tissue attenuation components and a few septa. The tumor displaced liver’s right lobe anteriorly and right kidney caudally, while the right adrenal gland was not identified in its normal anatomical position (Figure We decided to proceed with tumor’s surgical removal after obtaining informed written consent from the patient. Our approach was laparoscopic lateral transperitoneal, under general anesthesia. A three-dimensional high-definition camera was used, and the configuration and numbering of trocars are demonstrated in Figure Macroscopic examination revealed a gray-yellow, encapsulated, irregularly shaped mass, 16.5 x 15 x 6.5 cm in size Figure . On cut The patient had an uneventful postoperative course and was discharged on the third postoperative day. The patient is followed up on an outpatient basis, and she demonstrated no recurrence on imaging six months postoperatively.AMLs are rare, benign tumors composed of mature adipose tissue and bone marrow represented by all three lineages of hematopoietic elements ,2. TheirThe origin and pathogenesis of AMLs are unclear ,3. The rAMLs are usually asymptomatic and incidentally identified during imaging studies presented for other reasons, as they are usually small, located retroperitoneally and hormonally inactive ,2. LargeAs observed in our case, AMLs are presented as hyperechoic or hypoechoic images on U/S, depending on the predominance of fatty tissue or bone marrow elements, respectively . On CT s111indium chloride bone marrow scan has been proposed as an adjunct in preoperative evaluation in such cases. In our case, we decided to approach the tumor as benign, based on preoperative imaging studies which demonstrated a lipid-rich, well-defined, encapsulated mass, which did not infiltrate or change the borders of surrounding tissues. The endoscopic appearance of the tumor during laparoscopy and the fact that it was easily dissected reinforced our initial planning and we did not proceed with wide resection, which is demanded in case of retroperitoneal liposarcoma [Lesions like adrenal adenoma or carcinoma, metastases, pheochromocytoma and renal angiomyelolipoma are usually excluded based on preoperative investigation . Extrameosarcoma . FinallyAMLs demonstrate no malignant potential, and active surveillance with regular imaging follow-up is the appropriate treatment for small, asymptomatic lesions . SurgicaPreoperative diagnosis of giant AML can be difficult as it resembles retroperitoneal liposarcoma. However, the surgical approach between these clinical entities differs significantly. The macroscopic appearance of the tumor during laparoscopy guided our decision to avoid wide resection and the final histological findings were concordant with the intraoperative ones. Giant AMLs can be surgically removed efficiently and safely by using the laparoscopic approach.


Less than 4% of melanomas are of mucosal origin, with primary anorectal mucosal melanomas comprising a small subset. Mucosal melanomas are often diagnosed at an advanced stage due to delay in patient presentation and obscured tumor origins leading to a more aggressive behavior and less favorable prognosis when compared with cutaneous melanomas. We present a case of metastatic anorectal mucosal melanoma with a negative colonoscopy 1 year earlier. Mucosal melanomas are rare and arise primarily in the head and neck, anorectal, and vulvovaginal regions. Anorectal mucosal melanoma (AMM) is an extremely rare entity, and there is a significant paucity of literature on this malignancy.4 AMM most often affects females in the fifth to seventh decade of life, and rectal bleeding is the most common chief complaint.5 AMM more often affects the rectum than the anal canal; however, the 2 entities usually are studied together.6 The significance of mucosal melanomas when compared with cutaneous melanomas lies in their propensity to be more aggressive with less favorable outcomes.4 Approximately 20%-70% of patients with AMM present with metastases at the time of diagnosis, with a dismal 5-year survival rate of 10%-20%.7 We present a case of AMM diagnosed by retroflexion maneuver performed during repeat colonoscopy in a female patient presenting with vague gastrointestinal symptoms with a normal digital rectal examination (DRE) and a normal colonoscopy within the past year.Colonoscopy is a highly sensitive and specific screening modality for colorectal cancer detection.A 68-year-old woman presented to the hospital with a history of several months of intermittent fecal impaction complicated by sporadic outlet type rectal bleeding, a negative screening colonoscopy 1 year earlier, and a normal DRE 2 months earlier. She endorsed progressive anorexia and a 20lb weight loss over the past few months. On presentation, she was hemodynamically stable with a normal hemoglobin level. Computed tomography imaging depicted multiple lesions in the liver and lungs concerning for metastatic disease.During repeat colonoscopy, a rigid mass was felt on rectal examination and granular nodularity with ulceration in the rectum extending up to the dentate line was visible on retroflexion Figure . A biopsThe patient was diagnosed with T4N1M1, stage IV AMM (BRAF/C-KIT negative), and was recommended to undergo ipilimumab and nivolumab (I3/N1) immunotherapy with palliative radiation. The patient completed 4 cycles of ipilimumab/nivolumab and a total of 2000 Gy in 5 fractions of radiation therapy without complication. Repeat imaging demonstrated a reduction in primary tumor and metastatic disease. She was continued on monthly maintenance nivolumab therapy. Patient still endorses symptoms of constipation with resolution of rectal bleeding.8 Despite these setbacks, a good-quality colonoscopic examination is imperative in diagnostic evaluation of this entity.Anorectal mucosal melanoma is a rare malignancy with a known poor prognosis. AMM's less favorable prognosis is believed to be related to delay in diagnosis because of late symptom presentation and from obscured tumor origins.8 In addition, retroflexion in the rectum serves a fundamental purpose in adenoma detection with more than 50% of all lesions in the distal rectum only being identified on retroflexion.9 The importance of these key steps in clinical evaluation of a patient presenting with unknown rectal bleeding is highlighted in this case report.A standardized, high-quality colonoscopic evaluation includes a DRE and retroflexion before endoscope withdrawal. DRE is an underused physical examination maneuver both inside of and outside of the endoscopy suite that can be used to assess not only for anorectal masses but also perineal sensation, anorectal function, and rectoceles.6 In addition, on immunohistochemistry, AMM normally shows a strong positivity to S100, Melan-A, tyrosinase, and HMB-45, while usually negative for pan-cytokeratin.6 Once diagnosed, treatment options for AMM are only moderately successful with surgery being the cornerstone of treatment. Surgical options include wide local excision (WLE) or radical excision ; however, there are no significant differences in survival between WLE and APR.11 WLE offers a much less morbid operation, while APR may offer a higher rate of local control.10 Finally, rate of locoregional recurrence is improved in APR (42%) compared with WLE (71%).10 Survival after surgical intervention significantly decreases with advanced staging. In addition to surgical intervention, treatment for mucosal melanoma includes c-KIT(imatinib), CTLA-4 (ipilimumab), and PD-1 inhibition (nivolumab) therapies.11 The objective response rate (ORR) is superior for PD-1 inhibition (ORR 35%) over CTLA-4 inhibition (ORR 10%) as single agents for mucosal melanoma; moreover, combination therapy has had promising response rates for cutaneous melanoma to be considered for mucosal melanoma.12Diagnosis of AMM is difficult with endoscopic appearance demonstrating ulcerated polypoid lesions with irregular surfaces and generally amelanotic but sometimes with brown/black pigmentation.10 High-quality colonoscopy is imperative in diagnosing this malignancy with strict adherence to standardized endoscopic evaluation protocols. This case is a prime example of not only the stealth and aggressive nature of anorectal mucosal melanoma presenting with disseminated disease despite normal colonoscopy 1 year earlier but also of the importance of the rectal examination and careful endoscopic retroflexion maneuver during colonoscopy.AMM is a deadly diagnosis that portends a poor prognosis because of it frequently being diagnosed late as advanced metastatic disease.Author contributions: V. Chittajallu wrote the manuscript and reviewed the literature. CR Simons revised the manuscript for intellectual content and provided the figures. O. Oshilaja provided the figures. P. Chahal revised the manuscript for intellectual content and is the article guarantor.Financial disclosure: None to report.Previous presentation: This case was presented at the Ohio Gastroenterology Society Annual Meeting, September 14, 2019; and the American College of Gastroenterology Annual Scientific Meeting, October 25-30, 2019; San Antonio, Texas.Informed consent was obtained for this case report.


Abdominal cocoon syndrome (ACS), also called sclerosing encapsulated peritonitis, is a condition characterized by encapsulation of all or some of small bowel loops by a thick fibrous membrane. Etiologic cause is not fully known. It is among the rare causes of intestinal obstruction in adults. Preoperative diagnosis is difficult, and high suspicion is required. Diagnosis is generally made during laparotomy performed due to mechanical obstruction. In treatment of the condition, large scale surgical resections should be avoided. In the present study, we aimed to evaluate all clinical and radiological characteristics and surgical treatment of ACS in light of the literature through four patients operated in our clinic. Abdominal cocoon syndrome (ACS) is a rare condition causing acute and subacute intestinal obstruction (IO) symptoms. It is caused by a complete or incomplete encapsulation of small bowel loops by a thick fibrous membrane. Manifestation of clinical symptoms is dependent upon the extent of fibrous capsule , 2. It iThe aim of the present study was to evaluate the clinical and radiological findings of four patients diagnosed with ACS who were operated with mechanic IO prediagnosis in light of the literature.A 30-year-old male patient with nausea, vomiting, and abdominal pain complaints lasting for a few days presented to our clinic. In his anamnesis, he had several previous abdominal pain attacks which healed themselves. He had not had any abdominal surgery or medicine use before. Clinical examination revealed a tender and distended abdomen. Plain X-ray of the abdomen showed air-fluid levels of small bowel type. On the other hand, abdominal CT revealed dilatation and wall thickness in the terminal ileum . No abnoA 47-year-old male patient presented to our hospital with the complaints of constipation, nausea, and vomiting for three days. No significant medical or clinical record was apparent in his medical past, including any previous abdominal operations or medication use. Clinical examination revealed signs of only abdominal distension. Plain abdominal X-ray indicated air-fluid levels while abdominal CT showed proximally dilated clustered terminal ileum and cecum. Laboratory parameters were normal, and exploratory laparotomy was carried out. During laparotomy, a cocoon-like fibrotic tissue with a diameter of about 10 cm was found to surround internally herniated ileum and cecum . Bowel lth postoperative day.A thirty-eight-year-old female patient applied to our hospital with the complaints of swelling, vomiting, and constipation for a week. She had sigmoid colon tumor diagnosis two months ago and underwent left hemicolectomy. Clinical examination revealed only distended abdomen. Plain X-ray of the abdomen indicated air-fluid levels while abdominal CT showed clustering of proximally dilated ileum and cecum. Laboratory findings were normal Figures . Explorath postoperative day.A sixty-four-year-old female patient applied to our hospital with the complaints of abdominal swelling, vomiting, and constipation which had been continuing for three days. Her history revealed TAH+BSO+pelvic and para-aortic lymph node dissection due to endometrium cancer a month ago. Clinical examination showed only abdominal distension. No feature was observed in plain abdominal X-ray and abdominal CT examinations. Laboratory parameters were normal Figures . ExploraACS, also known as abdominal cocoon, sclerosing encapsulated peritonitis, primer sclerosing peritonitis, or idiopathic sclerosing peritonitis, was first described by Hu et al. and Kanat et al. , 4. It iSome publications categorize ACS in three classes based on the affected organ. In type 1 ACS, a small section of small bowel is surrounded by fibrous capsule. In type 2, all of small bowels are encapsulated by fibrous capsule, and in type 3, all small bowels and other organs are encapsulated by fibrous capsule , 11. ACSACS is a condition in which small bowel is partly or completely enclosed by a fibrous capsule in adults. It is among the rare causes of mechanical intestinal obstruction. The disease could develop with acute or subacute manifestations. Preoperative diagnosis is difficult, and diagnosis is generally made during laparotomy. In patients with incomplete type of ACS, it can be possible to achieve complete healing. On the other hand, short-term mortality is common in the cases with complete type of the disease. ACS should be considered in the differential diagnosis of patients who presented with acute or subacute ileus attacks.


Mixed gestational trophoblastic neoplasms are extremely rare and comprise a group of fetal trophoblastic tumors including choriocarcinomas, epithelioid trophoblastic tumors, and placental site trophoblastic tumors. We present a case of a patient with extrauterine mixed gestational trophoblastic neoplasm adjacent to the abdominal wall cesarean scar. On the basis of a literature review, this type of case has never been reported before due to the unique lesion location and low incidence.Our patient was a 39-year-old Chinese woman who had a history of two cesarean sections and one miscarriage. She had a recurrent anterior abdominal wall mass around her cesarean scar, and the mass was initially suspected of being choriocarcinoma of unknown origin. The patient had concomitant negative or mildly increased serum β-human chorionic gonadotropin at follow-up and no abnormal vaginal bleeding or abdominal pain. However, she underwent local excision twice and had two courses of chemotherapy with an etoposide and cisplatin regimen. She finally opted for exploratory laparotomy with abdominal wall lesion removal, subtotal hysterectomy, bilateral salpingectomy, and left ovarian cyst resection, which showed the abdominal wall lesion, whose components were revealed by microscopy and immunohistochemical staining to be approximately 90% epithelioid trophoblastic tumors and 10% choriocarcinomas from a solely extrauterine mixed gestational trophoblastic neoplasm around an abdominal wall cesarean scar.It is worth noting whether epithelioid trophoblastic tumor exists in the setting of persistent positive low-level β-human chorionic gonadotropin. More studies are required to provide mechanistic insights into these mixed gestational trophoblastic neoplasms. Gestational trophoblastic neoplasms (GTNs) are a group of malignant fetal trophoblastic tumors that include choriocarcinomas (CCs), placental site trophoblastic tumors (PSTTs), and epithelioid trophoblastic tumors (ETTs). CCs are composed of variable proportions of neoplastic cytotrophoblasts, syncytiotrophoblasts, and intermediate trophoblasts. Intermediate trophoblastic lesions include exaggerated placental sites, placental site nodules (PSNs), PSTTs, and ETTs .However, the exact pathogenesis of the differentiation of GTNs, especially the mixed GTNs, is still unknown because these tumors are extremely rare . To our Our patient was a 39-year-old Chinese woman who had delivered a second live full-term infant through an uncomplicated cesarean section in 2011 after a miscarriage in 2010 and a first cesarean section in 2005. In December 2014, she noticed a purple, nontender swelling appearing as an anterior abdominal wall mass around her cesarean scar. The mass was the size of a green bean and was not accompanied by any abdominal pain or abnormal vaginal bleeding. There were no notable findings in her past medical history, family history, or psychosocial history. The abdominal wall mass had progressively enlarged, which led to her presentation to a local institution. She had undergone tumor resection of the abdominal wall in June 2015, which might be interpreted as the abdominal wall endometriosis malignancy.The patient had visited a regional tertiary hospital for a consultation regarding the pathologic diagnosis of a CC before presenting to our hospital for further diagnosis and treatment. Her physical examination showed no abnormalities except for the scar from the local excision on the abdominal wall. Our pathologists reviewed the first excised specimen from the previous hospital and confirmed the characteristic of CC coexisting with minor ETT. Her Ki-67 proliferative index was approximately 50%. Laboratory analysis revealed normal serum levels of β-human chorionic gonadotropin  and tumor markers, including carbohydrate antigen 125 (CA 125), carcinoembryonic antigen (CEA), CA 19-9, CA 15-3, and α-fetoprotein; all of these biomarkers had consistently negative values. Positron emission tomography was performed to further determine whether other metastatic lesions existed; however, no residual tumor and suspicious malignant lesions were observed. For further evaluation, endometrial curettage was performed, the results of which revealed normal menstrual phase endometrium. Subsequently, the patient received two courses of chemotherapy with a regimen of etoposide and cisplatin (EP) over a 2-month period. During chemotherapy, her serum β-hCG levels remained negative (< 1.2 IU/L).Subsequently, she underwent regular follow-up in the outpatient department, and a recurrent nodule was found on the same abdominal wall scar site in January 2017, approximately 17 months after the last chemotherapy. The patient was registered for admission again. Her serum β-hCG had increased to 6.17 IU/L, and two oval hypoechoic masses were visualized by ultrasonography in the subcutaneous soft tissue of the lower abdomen wall scar. Chest computed tomography (CT) and head magnetic resonance imaging (MRI) showed no abnormality. Then she underwent a second notable mass excision by ultrasound interventional localization. In this case, the nodule was in the fat layer next to the superficial fascia. Her serum β-hCG level was decreased to 3.4 IU/L on the second postoperative day. The result of pathological examination was initially in line with CC metastasis to the abdominal wall, and the patient’s Ki-67 index was 20%. However, the patient’s pathological sections were sent to the Fudan University Obstetrics and Gynecology Hospital, another tertiary hospital in China, for further consultation, and the finding was ETT. Finally, the patient was encouraged to maintain close follow-up, and her serum β-hCG level had gradually decreased.In the follow-up visits, the patient’s serum β-hCG level was elevated to the highest level of 10.68 IU/L again 4 months later Fig. a and b. Histopathological observations suggested that hyperplasia of fatty fibrous tissue was visible with cancer infiltration and necrosis around the cancer tissue, which was consistent with trophoblastic tumor, constituted primarily by major epidermoid trophoblastic tumors (approximately 90%) and the remainder by CC components (approximately 10%) on the abdominal wall lesion Fig. a. ImmunoMixed GTNs are extremely rare, with only 20 mixed trophoblastic tumors having been reported , includiet al. [Mixed GTNs have clinical features more similar to those of intermediate trophoblastic tumors , 15. GTNet al. , becauseet al. reported [The distinction of ETT from other trophoblastic tumors is more challenging. Unlike CCs and hydatidiform moles, the serum β-hCG level with ETT is usually within the normal range or is slightly elevated, as Palmer reported . Our patreported . Patholoreported . Immunosreported . More imreported , 25. Howet al., CC was the most primitive trophoblastic tumor, whereas PSTT or ETT was relatively more differentiated [The pathogenesis of mixed trophoblastic tumors is not well known. According to the model presented by Mao entiated . Furtherentiated . This isentiated . Howeverentiated . These fentiated .After our patient presented to the hospital, she received two chemotherapy regimens with EP after the first local resection of the abdominal wall mass. Our pathologists had made a mistake in interpreting the second surgical specimen; other pathologists from different tertiary hospitals realized it was an ETT, not a CC, upon analyzing the same specimen. With the third recurrence, the final pathologic diagnosis was predominantly ETT coexisting with a small proportion of CC that was only present on the abdominal wall lesion. On the basis of the above information, we could not rule out the possibility that the predominant ETT lesion might be associated with the previous CC. However, there is still not enough evidence to support the notion that the predominant ETT was a result of previous chemotherapy.Currently, there is extremely limited experience with ETT coexisting with CC. CC is sensitive to chemotherapeutics, but PSTT and ETT are not. Total hysterectomy with lymph node dissection was recommended for mixed GTNs, and chemotherapy should be used in patients with metastatic disease or in patients without metastatic disease but with adverse prognostic factors . FurtherMixed gestational trophoblastic tumors are rare. We report the first case of extrauterine mixed trophoblastic tumor around an abdominal wall cesarean scar. It is essential to distinguish ETT from PSTT, CC, and other malignancies, especially in normal tissue in the uterus. Pathologists and clinicians should pay more attention to mutual communication and arrive at a correct diagnosis and finally select an optimal therapeutic schedule. Because limited data are available related to mixed GTNs, long follow-up is necessary, and additional cases must be accumulated.


Infective endocarditis and cardiac myxoma have common features including fever, systemic embolism and intra-cardiac masses. For this reason, these diseases are often misdiagnosed one for another despite proper imaging studies. Herein, we report a case of suspected infective endocarditis in a patient with acute stroke, fever and a mass adjacent to the mitral valve.A 24-year-old male patient presented with recurrent fever and stroke. In view of a history of Cushing syndrome and a mobile mass in the left atrium, infective endocarditis was highly suspected. He was transferred for emergency cardiac surgical intervention. During surgery, intraoperative transesophageal echocardiography revealed a 7 cm mass attached to the interatrial septum. The mass was excised through right mini-thoracotomy and pathological examination confirmed the presence of a myxoma. Based on the above clinical findings and genetic analysis, the diagnosis of Carney complex was confirmed.Infective endocarditis and cardiac myxoma have common features and can be misdiagnosed. If a young patient presenting with embolic stroke had a history of an endocrine neoplasm, Carney complex should be considered in the differential diagnosis of infective endocarditis. At least 20% of all ischemic strokes originate in the heart. Especially, young patients presenting with acute stroke should be evaluated for an intra-cardiac source of embolism. Among them, infective endocarditis (IE) and cardiac myxoma (CM) share common features such as fever, systemic embolism, and an intra-cardiac mass. A recent publication also showed a case of CM initially misdiagnosed as IE . These dA 24-year old male patient visited the emergency department of a tertiary hospital for increased drowsiness and fever. The patient had a history of Cushing’s syndrome Fig. a and hadPPKAR1A gene. Therefore, he was finally diagnosed with CNC presenting with CM and systemic embolism.During surgery, intraoperative transesophageal echocardiography (TEE) revealed that the mass was not attached to the mitral valve but to the interatrial septum Fig. a. The maIn the present case, symptoms and signs such as skin striae in the torso, relapsing fever, a highly mobile mass on the mitral valve, and embolic cerebral infarction led to a high level of suspicion for IE. Additionally, the immunocompromised state due to long-term steroid replacement therapy supported the probability of IE. However, intraoperative TEE helped confirm that the mass was attached to the interatrial septum and not to the mitral valve, thereby showing the mass to be a myxoma. CM is the most frequently occurring benign primary cardiac tumor, accounting for 0.3% of open-heart surgeries . While vMost cases of CM are benign and sporadic. Familial forms of myxoma also exist and typically occur in young, male patients in unusual locations, and have a tendency to be multifocal . CNC is In the present case, the precipitous treatment with intravenous tPA and mechanical thrombectomy delayed surgery. For young patients presenting with stroke, prompt evaluation of the source of embolism should be done prior to stroke treatment. Echocardiography and computed tomography can provide valuable information to help establish a treatment plan and minimize the possibility of additional embolic events.In summary, infective endocarditis might be suspected in patients with a history of recurrent fever, stroke, and a mobile mass in the left atrium. However, due to similarities in symptoms and signs, Carney complex associated cardiac myxoma should also be included in the differential diagnosis, especially in case of young patients with a history of endocrine neoplasms.


Pancreatic serous cystadenoma (SCA) is a benign, cystic lesion with an indolent growth pattern. Complications such as spontaneous hemorrhage or malignant transformation from SCA are extremely rare. Our case report describes an unusual presentation of a patient with a previously diagnosed SCA, made unique by the presence of three separate neoplasms in the final specimen.A 74-year-old male with a previous diagnosis of SCA presented emergently with epigastric pain and non-bilious vomiting. Laboratory results were notable for a hemoglobin of 8.3 g/dl. CT scan of the abdomen demonstrated a complex, solid-cystic mass in the pancreatic head with a large hematoma and questionable focus of active hemorrhage. Surgical resection was recommended due to the risk of malignancy, possibility of re-bleeding, and symptoms of severe duodenal compression. Pancreaticoduodenectomy was performed, and final pathology demonstrated three separate neoplasms: serous cystadenoma, intraductal papillary mucinous neoplasm, and neuroendocrine tumor.While pancreatic SCA are benign tumors that can be observed safely in the majority of cases, surgical intervention is often indicated in patients with large, symptomatic cysts or when diagnosis is unclear. When undergoing surveillance, it is crucial for both the patient and the care team to be aware of the possibility of rare, but life-threatening complications, such as hemorrhage. Likewise, the possibility of misdiagnosis or concurrent neoplasia should be considered. Pancreatic cystic neoplasm (PCN) represents a diverse group of pathologic entities with a wide range of clinical implications—most commonly, serous cystadenoma (SCA), mucinous cystic neoplasm (MCN), intraductal papillary mucinous neoplasm (IPMN), cystic pancreatic neuroendocrine tumor (pNET), and solid pseudopapillary neoplasm (SPEN) . PCN is Observation has been favored historically once SCA is diagnosed, as the malignant potential is extremely low. Asymptomatic patients with SCA usually undergo surveillance with serial imaging , and resWe present a rare case of a patient who presented with spontaneous hemorrhage from a large cystic neoplasm in the pancreatic head diagnosed as SCA 5 years earlier. The patient underwent pancreaticoduodenectomy, and the final pathology was remarkable for three separate neoplasms: SCA, IPMN, and pNET. We discuss the risks posed by SCA, as well as the importance of structured surveillance once SCA is suspected.A 74-year-old male, with a past medical history of SCA diagnosed 5 years prior to presentation, hypertension, nephrolithiasis, and childhood polio with consequent bilateral lower extremity atrophy, presented to the emergency department with epigastric pain associated with persistent, non-bilious vomiting. Patient mentioned a history of a pancreatic cyst, which he had been told was harmless. A CT scan from 5 years prior to his presentation demonstrated a 10 × 8 cm septated cystic lesion in the head of the pancreas  surgery team for a Whipple procedure on hospital day 3. Intraoperatively, a large hematoma was found adjacent to the lesion, extending posterolaterally from the second portion of the duodenum compressing the duodenal lumen. There was no evidence of vascular invasion.Pathology from the specimen demonstrated chronically inflamed pancreatic tissue and three separate neoplasms. A main duct IMPN Fig. a was seeThe postoperative course was uneventful. Patient was discharged on postoperative day 11, delayed due to the patient’s need for extensive physical therapy given pre-existing conditions. The patient has since recovered from the procedure and is doing well. Given the absence of invasive carcinoma, no adjuvant therapy was offered. Surveillance imaging is scheduled 6 months postoperatively.SCAs are benign tumors that exhibit an indolent growth pattern. They are more commonly found in women, with a median age of 58 years at diagnosis . Many arGiven the benign natural history of SCA, observation of asymptomatic patients is typically recommended. There is currently no consensus on management strategies, with recommendations ranging from surveillance for asymptomatic patients to surgical resection depending on various factors such as SCA size, location, or growth rates , 4, 10. Endoscopic ultrasound (EUS) can provide supplemental information, especially if fine needle aspiration (FNA) is performed for fluid analysis. Cyst fluid carcinoembryonic antigen (CEA) level has been useful in differentiating between mucinous and non-mucinous pancreatic cysts . VasculaEven when a clear diagnosis of SCA is made based on radiographic and clinical findings, different management strategies have been recommended. The European Study Group on cystic tumors of the pancreas recommends observation for 1 year for asymptomatic patients followed by symptom-based follow-up and surgical resection for patients with symptoms related to compression of adjacent organs . AnotherOur case report describes a patient who developed spontaneous hemorrhage associated with previously diagnosed SCA. Only a few cases of hemorrhage related to SCA have been reported based on our literature search. In two case reports, patients presented with acute abdomen from hemorrhage and underwent distal pancreatectomy of large cysts with a diameter > 10 cm , 8. WhilOur case report is also unique because the final pathology remarkably identified three different types of neoplasm: macrocytic serous cystadenoma, main duct IPMN, and well-differentiated neuroendocrine tumor. SCAs have been found concurrently with one other type of pancreatic tumors within the same excised specimen, such as IPMN, pancreatic ductal adenocarcinomas, neuroendocrine tumors, and metastatic tumors. In a study of 193 pancreatic serous neoplasms, concurrent neoplasms were found in 13% (27) of cases [We report the first case of SCA with two synchronous pancreatic neoplasms. While pancreatic SCA are typically benign tumors, the possibility of misdiagnosis or concurrent neoplasia should be considered. When opting for observation, it is crucial for both the patient and the care team to be aware that the tumor may grow, causing local mass effect and eventual symptoms, and that life-threatening complications may arise in rare cases.


Colo-rectal intussusception is rare in adults and is often secondary to malignant lesions, rarely benign lesions such as colonic lipomas can also be the cause. We present the case a 60-year-old man who presented to the emergency department with acute abdominal pain. On physical examination, the abdomen was distended with diffuse tenderness. CT scan of the abdomen revealed a colo-rectal intussusception secondary to a rectal lipoma with parietal pneumatosis of the invaginated loop. An emergency laparotomy was performed. Intraoperatively the radiological findings were confirmed. A rectosigmoid resection (Hartmann's procedure) taking off the lipoma and the invaginated segment of the colon was performed and the patient had an unevent full recovery. Histopathology confirmed a 6cm sub-mucosal lipoma without evidence of malignancy. As the diagnosis of a benign disease in patients presenting with colonic intussusception can only be made on pathological examination, this entity should be managed as a malignant lesion due to the high incidence of malignancy. Intussusception is a rare clinical entity in adults, defined as the telescoping of a proximal segment of a gastrointestinal tract intoan adjacent distal segment . It accoA 60-year-old man, with no medical or surgical history, presented to the emergency department with acute abdominal pain. He reported a short history of constipation and intermittent bleeding through the rectum for the last two weeks. On physical examination, vital signs were normal. The patient was afebrile. The abdomen was distended with diffuse tenderness. Furthermore, no palpable abdominal mass was detected. Rectal examination revealed a smooth and well-circumscribed mass lying in 3cm from the anal verge. Laboratory investigations on admission were normal except for a high level of weight blood cells (WBC = 19000 C/ml) and C-reactive protein (CRP = 170 mg/l). Computed tomography scan (CT) of the abdomen showed a 6cm soft-tissue mass with fat density within the rectum likely to be the lead point for a colorectal intussusception . Large bIntussusception is more commonly seen in pediatric patients and represents less than 5% of intestinal obstructions in adult . The meaLipoma is a begnin tumor of a mesenchymal origin, it occurs more frequently in the large intestine, mostly in the right colon . The incAbdominal computed tomography (CT) scan is the most specific diagnostic test for intussusception and is superior to ultrasonography and endoscopy . Its senSigmoido-rectal intussusception in adults secondary to a colonic lipoma is rare. The diagnosis of intussusception in adults is challenging because its non specific symptoms. CT scan has been reported to be the most specific radiological investigation for the diagnosis of intussusception especially in acute forms. Surgical resection, is recommended because of the high probability of malignancy.


When it comes to tumors of the head and neck, including NHL, the larynx should also be considered as a site for a possible secondary as well as primary involvement.The secondary manifestation of a marginal zone lymphoma (MZL), which is a less common type of B-cell non-Hodgkin’s Lymphoma (NHL), in the larynx is a rarity. We report a case of the secondary involvement of the larynx following MZL in a 72-year-old woman who presented with the sensation of a foreign body in the throat and history of MZL. A fiberoptic laryngoscopy confirmed the presence of a mass in the supraglottic area. The initial clinical evaluation indicated that the mass was benign. Hence, it was removed surgically. However, the histopathological analysis confirmed the diagnosis of MZL. After a systemic evaluation, the patient was classified as stage IV A according to the Ann Arbor staging system for lymphomas. Treatment was initiated with Ibrutinib 520mg/d and Rituximab 375 mg/m NHL of the head and neck can be divided into primary and secondary in terms of the original location of the lymphoma. The manifestation of NHL can be further divided into nodal, lymphatic extranodal , and extra lymphatic extranodal (orbit and larynx) in terms of the affected tissues. The extralymphatic, extranodal manifestation is an uncommon finding in this region -4.The differentiation of primary from secondary NHL of the larynx is essential for therapy decisions. Local radiotherapy is the mainstay for primary involvement, whereas secondary involvement indicates a higher stage and mandates systemic chemotherapy and immunotherapy , 6.A 72-year-old woman was referred to our department because of the sensation of a foreign body in her throat. She denied dysphagia, dyspnea, and B symptoms. She had a history of stage IVA MZL with evidence of IgG kappa monoclonal gammopathy, which was diagnosed 17 years ago. She received chlorambucil and prednisolone as therapy at the time. While receiving that therapy she developed a further orbital involvement of her lymphoma which was managed with local irradiation of the orbit with a total dose of 46.8 Gray. The patient obtained six courses of rituximab and bendamustin. Through the course of the therapy, she developed therapy-attributed myelodysplastic syndrome (t-MDS).After the therapy, she expressed a stable disease status and was kept under the watchful waiting approach till the latest presentation. She also reported a history of idiopathic thrombocytopenic purpura (ITP) and arterial hypertension.A fiberoptic laryngoscopy revealed a 2.5 x 2.5 cm round non-ulcerated mass arising from the left aryepiglottic fold in the supraglottic region. The mass was suspected to be a benign endolaryngeal polyp. No other pathological findings were detected. Due to technical issues on the day of the presentation, fiberoptic images could not be obtained during the laryngoscopy.Laboratory blood tests revealed her lactate dehydrogenase value to be 397 U/L and pancytopenia.Cancer staging with a full-body CT scan was performed six months prior to the presentation Figure . During Since the endolaryngeal mass was suspected to be a polyp and the secondary MZL involvement of the larynx was deemed highly unlikely, an avoidable surgery was planned, and unfortunately, no PET-CT imaging was conducted.The patient underwent microlaryngoscopic surgery under general anesthesia for resection and evaluation of the mass. Histopathological analyses were performed on the formalin-fixed, paraffin-embedded tissue sections of the resected mass. Lymphoid infiltration among normal epithelial cells was identified. Immunohistochemical staining showed a strong expression of CD20(+), Bcl6(-), CD10(-). Light chain Kappa restriction, about 20% CD138 (+) plasmacytoid cells, and proliferation fraction of about 20% were detected in the tumor cells. Molecular genetic analyses of the resected mass did not identify a mutation for Myd88 (exon 5). Based on the histopathological and molecular histological findings a diagnosis of marginal zone NHL was confirmed.For staging the patient underwent a bone marrow biopsy. The cytological and histological analyses showed no infiltration of the bone marrow with lymphoma but revealed a secondary acute myeloid leukemia (s-AML) phenotype M6. The s-AML developed secondary to the therapy (t-MDS). A contrast-enhanced full-body CT scan (compared to the CT scan performed six months earlier) revealed progressive lymph nodes in the posterior mediastinum which had been in partial remission previously. There was no evidence of the involvement of other lymph node groups. A previously known slight splenomegaly was stable. The CT scan shows no remnant of the initial endolaryngeal mass Figure . LaboratThe patient was referred to the Department of Hematology and Oncology for continued management. Treatment was initiated with ibrutinib 520mg/d and rituximab 375 mg/m2 every 28 days.Six and 12 months follow-ups with fiberoptic laryngoscopy showed no local recurrence of the endolaryngeal mass. A full-body CT scan showed a stable disease status.The majority of secondary malignant involvements of the larynx arise from breast cancer or malignant lymphatic proliferation . A seconMZL is a less common type of B-NHL disease and contributes to 5-15% of all lymphomas in total. However, a retrospective study of 200 patients diagnosed with laryngeal lymphomas between 1973 and 2014 found interestingly that 16.5% of laryngeal lymphoma were MZL [The most common extranodal localizations of primary MZL are the stomach, followed by the adnexa and the orbit , 4. LessThe clinical features of our case are consistent with similar reported cases regarding presentation and clinical findings. There are no means to differentiate between primary and secondary involvement based only on clinical examination , 7. In oWhile most of the published cases are about the primary involvement of the larynx with NHL, which indicates Stage IE according to the Ann Arbor staging system for lymphomas, our case describes a secondary involvement of the larynx with MZL which indicates Stage IV A . NonetheThe differentiation between primary and secondary larynx involvement of MZL is paramount as it affects therapy regimes and prognosis . PrimaryIn patients with MZL, it is paramount to thoroughly inspect uncommon sites such as the larynx for secondary involvement by radiological imaging. Misdiagnosing such an involvement can falsely downgrade the stage of the disease and delay proper treatment. Moreover, it may expose the patients to unnecessary surgical procedures. As benign endolaryngeal masses may mimic the MZL of the larynx clinically, a PET-CT scan before proceeding with the surgical planning is very helpful in differentiating MZL of the larynx from benign endolaryngeal lesions.It is essential to differentiate primary from secondary involvement of the larynx in cases of malignant lymphatic proliferation as it has a great impact on further management, therapy, and prognosis. While local irradiation is the main therapy for primary involvement of the larynx, secondary involvement of the larynx in a disseminated disease may require a combination of systemic chemotherapy and immunotherapy.


Malignant melanoma (MM) generally presents as a primary neoplasm of the skin, and most MM cases of the respiratory system are metastatic. Primary MM of the lung (PMML) is quite rare, and its diagnosis is relatively difficult.We report the case of a 57-year-old male patient with PMML who denied any history of tumours. His initial complaint was frequent coughs with bloody sputum for 4 days. Chest radiography demonstrated a high-density shadow in the lower lobe of the right lung, which was suspected to be a large space-occupying lesion on subsequent computed tomography (CT) and to be a hypermetabolic tumour by positron emission tomography–CT. To confirm the diagnosis, exploratory surgery was performed. Finally, we confirmed the diagnosis of PMML.PMML is extremely rare and easily misdiagnosed as lung cancer. Because of its morphological and immunophenotypic variations, the diagnosis of PMML remains difficult. This case report discusses the diagnosis and case management of a patient while referring to the existing literature. Malignant melanoma (MM) is a refractory malignant tumour. In 2015, approximately 351,880 new cases of MM were diagnosed worldwide [A 57-year-old man presented to Jinhua Central Hospital affiliated with Zhejiang University  with a complaint of frequent cough with bloody sputum for 4 days. He denied any history of skin, mucous membrane or eye surgeries; electric cauterisation; or any family history of cancer. Physical examination at the outpatient clinic revealed diminished breath sounds over the lower lobe of the right lung, and no abnormal lesions were detected in other sites of the body, including the skin, head, neck, scalp, anogenital region and eyes. Chest radiography showed a high-density shadow. Subsequent CT plain and contrast-enhanced scan showed a space-occupying lesion in the lower lobe of the right lung adjacent to the pleura with a clear boundary. A right lower lobectomy for the space-occupying lesion was performed and further diagnosed by pathological examination. Before the excision, whole body positron emission tomography–CT (PET-CT) was performed, showing a malignant space-occupying lesion in the lower lobe of the right lung with liver metastasis Fig. .Fig. 1PGrossly, a solitary mass measuring 3.5 cm × 3.0 cm × 3.0 cm was located in the lower lobe of the right lung, appearing as a round, darkly pigmented, solid neoplasm with significant necrosis. Hematoxylin and eosin staining demonstrated that the tumour was located in the lung tissue, comprising malignant epithelial tumour cells with large amounts of acidophilic cytoplasm and prominent nuclei. Melanin pigmentation could also sometimes be noticed, and there were junctional changes with characteristic tumour cells invading the bronchial subepithelial area from the basement membrane. All of these findings were suggestive of MM Fig. . ImmunohBased on the clinical characteristics, CT and PET-CT findings and pathological evaluation, a final diagnosis of PMML was made. After surgery, the patient received one cycle of chemotherapy. However, as his general condition was very poor, the patient died of pulmonary infection 27 days later.PMML is extremely rare but characterised by high malignancy, a high recurrence rate and poor prognosis. Only about 47 cases have been reported in the literature , 4. The The diagnosis criteria of PMML have been clearly defined by Jensen and Egedorf , with onDuring the diagnosis, attention should be paid to distinguishing PMML from some similar tumours such as squamous cell carcinoma (SCC), pulmonary metastatic melanoma (PMM), and pigmented carcinoid tumour (PCT) –17. The The clinical presentation of PMML varies with individual cases. Yamamoto et al.  reportedIn summary, PMML is extremely rare and easily misdiagnosed as lung cancer. Histopathological examination is a reliable gold standard for confirmation of a PMML diagnosis. Because of the morphological and immunophenotypic variations, the diagnosis of PMML remains difficult and should be distinguished from the diagnosis of other cancers. Immunohistochemistry staining is helpful for the differential diagnosis of PMML. Because of the lack of clinical data and clear understanding of the mechanisms underlying the pathogenesis of PMML, there are no consensus guidelines on the treatment of PMML. We hope that the case reported here will add more clinical data to the current information about PMML and help to provide more constructive suggestions about the treatment of PMML.