Atypical carcinoid arising from the mediastinal tissue is a rare neuroendocrine tumor and an association with parathyroid adenoma is very unusual. We report an unusual case of atypical carcinoid of mediastinum with metastasis in a patient presenting with parathyroid adenoma, which is the first case to be reported from Pakistan.A 51-year-old Pakistani man was seen in postoperative intensive care after right parathyroidectomy and mediastinal mass resection for the management of postoperative hypocalcaemia. He had a background history of dyspnea. Examination was unremarkable. Preoperative laboratory evaluation revealed a calcium level of 12.7 mg/dl, phosphate of 1.9 mg/dl, serum albumin of 4.8 g/dl, alkaline phosphate of 94 U/L, and serum intact parathyroid hormone level 413.8 pg/ml. A technetium-99m sestamibi parathyroid scan showed right parathyroid increased tracer uptake. Further workup revealed a large mediastinal mass which was diagnosed as atypical carcinoid after Tru-Cut biopsy. He underwent right-sided parathyroidectomy and resection of the mediastinal mass. The histopathology confirmed it to be a parathyroid adenoma and atypical carcinoid tumor of his mediastinum with metastasis in his lymph node and parathyroid gland. Somatostatin receptor scintigraphy revealed a well-defined focus in his left hypochondriac region consistent with a somatostatin receptor scintigraphy-avid tumor. He was started on everolimus and planned for octreotide therapy. We describe an incidental finding of atypical carcinoid of the mediastinum in a patient diagnosed as having parathyroid adenoma. Clinical manifestations of neuroendocrine syndromes are challenging. Some tumors cluster in a non-classic description with other common neoplasms. They rarely present in isolation, remain clinically silent, and need aggressive workup with the aid of imaging and histopathology. Primary neuroendocrine tumors of the mediastinum are rare. They can arise from thymic or non-thymic tissue, can be low to high grade and classified as well-differentiated or poorly differentiated neoplasms . Among t4) 1.9 mg/dl, and serum albumin of 4.8g/dl; his alkaline phosphate 94 U/L and serum intact parathyroid hormone (PTH) level was 413.8 pg/ml. His serum creatinine was 1.04 mg/dl and he had an estimated glomerular filtration rate (eGFR) of 75.28 ml per minute/1.73m2. An ultrasound of his abdomen showed bilateral medullary nephrocalcinosis. An ultrasound of his neck revealed that his right thyroid lobe was mildly enlarged and both lobes showed a few small scattered solid nodules less than 10 mm. His right inferior parathyroid gland was markedly enlarged measuring 3.9×1.3×2.3 cm. Thyroid function tests were normal. A technetium-99m (99mTc) sestamibi parathyroid scan was carried out, which showed a focal area of intensely increased tracer uptake in his right thyroid lobe, which after washout still showed intense tracer accumulation with a probability of parathyroid adenoma or hyperplasia in the right group of parathyroid glands , sitagliptin, metformin, and furosemide. His family history was insignificant. On examination he was well oriented and his vital signs were stable. A general physical examination revealed only mild pedal edema. A systemic examination was unremarkable. Preoperative investigations showed a calcium (Ca) level of 12.7 mg/dl, phosphate . In some areas the tumor cells formed rosette-like structures. Large areas of necrosis were seen along with extensive homogenous, fibrotic areas Fig. . These aNeuroendocrine tumors arising from the thymus itself account for just 2 to 5% of primary thymic malignancies . AtypicaThere is a case report of atypical carcinoid originating from the thymus gland in the form of a mediastinal mass in association with hypercalcemia which was found to be due to parathyroid adenoma . AnotherWe presented a case of metastatic mediastinal atypical carcinoid in a patient with parathyroid adenoma, diagnosed incidentally during preoperative workup. The unusual clustering of neuroendocrine tumors is being increasingly reported in the literature, and patients with hyperparathyroidism should be thoroughly investigated to diagnose and treat other related tumors particularly atypical carcinoid of mediastinum as it has a poor outcome and limited treatment options.


Sarcoidosis is a multi-organ disease of unknown etiology characterised by the presence of epithelioid granulomas, without caseous necrosis. Systemic sarcoidosis is rare among children, while neurosarcoidosis in children is even rarer whether it is systemic or not.We described the case of a 12-year-old boy who presented with monocular vision loss accompanied by unusual MRI features of an extensive meningeal infiltrating mass lesion. The patient underwent surgical resection (biopsy) via a frontotemporal craniotomy to establish a definitive diagnosis based on the histopathology, since neurosarcoidosis remains a very difficult diagnosis to establish from neuroradiogenic imagings. Based on the histopathology of the resected mass lesion, neurosarcoidosis was diagnosed. On follow-up after 3 months of steroid therapy, the patient displayed a good response on the imaging studies. MRI revealed that the preexisting mass lesion had regressed extremely. We also conducted a small literature review on imaging studies, manifestations, appropriate treatments, etc., in particular neurosarcoidosis including children.Although extremely rare, neurosarcoidosis, even in children, should be considered in the differential diagnosis of skull base mass lesions to avoid unnecessary aggressive surgery and delay in treatment, since surgery may have little role in the treatment of sarcoidosis. Sarcoidosis is an idiopathic granulomatous disease which commonly involves the lungs, skin, and eyes, with simultaneous or metachronous features , 2. SarcWe described here the rare clinical and radiological manifestations of 12-year-old boy with monocular vision loss who was initially thought to have a skull base tumor, but was subsequently confirmed to have neurosarcoidosis with systemic sarcoidosis. In addition, we present a small literature review of neurosarcoidosis, especially in pediatric patients.A 12-year-old boy was introduced to our institution because he displayed visual disturbance and was described as having an intracranial mass lesion by another hospital. His family history was unremarkable. His past history included a right neck mass lesion: this had expanded to 40 mm in 1 month and was histologically suspected to be associated with sarcoidosis by another hospital 2 years ago. Subsequently, he was followed up at that hospital, but there was no obvious abnormality until 3 months ago when visual disturbance was identified at an annual school medical checkup.His consciousness level was clear and neurological examinations demonstrated no abnormalities except for the ophthalomological problem. Neurological examinations, including the I and III – XII cranial nerves, detected no abnormality. Ophthalomological evaluations revealed concentric contraction of the visual field and diminished visual acuity of the right eye. His best corrected visual acuity was 0.2/20 (n.c) in the right eye and 4/20 (1.2pxS-1.5D) in the left eye. The pupils were semidilated and the pupillary light reflex was sluggish on the right side, while a normal size and normal reaction were evident on the left side. In a fundus examination, his right optic disc was found to be pale.Routine laboratory tests including renal function and hormonal tests demonstrated no obvious abnormalities. As a marker for sarcoidosis, his serum angiotensin-converting enzyme (ACE) was slightly increased at 22.5 U/L (8.3 to 21.5 U/L). His chest X-ray performed on admission showed no obvious abnormalities, but a subsequent chest computed tomography (CT) scan revealed small scattered pulmonary nodules in the bilateral lungs.A CT scan and magnetic resonance imaging (MRI) of the head demonstrated that a leaf-shaped extra-axial mass of about 73 mm in length was present around the right cavernous sinus, straddling the sella turcica, frontal, middle, and posterior cranial fossa, with dural thickness to the tentorium cerebrii Figs. , 2, 3. TThe patient underwent surgical resection (biopsy) via a frontotemporal craniotomy to establish a definitive diagnosis based on the histopathology, since neurosarcoidosis remains a very difficult diagnosis to establish from neuroradiogenic imagings.The intraoperative findings indicated that the tumor was hard, and visually and tactually resembled like a meningioma. The lesion was found immediately after opening the sylvian fissure, and was easy to separate from the surrounding brain parenchyma in the frontal cortex, but had no distinct boundaries in the temporal cortex. When reaching deeply into the sylvian fissure, the internal cerebral artery was seen to be involved within the lesion. The immediate pathology revealed that the specimen consisted of a proliferation of short spindle cells without necrosis, with multinucleated giant cells. Surgical exploration was therefore terminated at this stage.The definitive histopathology of the resected mass lesion showed epithelioid cell granulomas and Langhans type multinucleated giant cells that were rich in dense fibrous tissue with lymphocytes and plasma cells. Necrosis was not clear in the granulomas observed in the specimen Fig. . GrocottBased on the histopathology of the resected mass lesion, neurosarcoidosis was diagnosed. Although steroid therapy was planned, the start was delayed due to epidural abscess of the surgical wound, which required a surgical procedure and antibacterial therapy. Three months after the biopsy, the patient was started on steroid therapy . Fortunately, during this delayed time, his neurological examinations did not change and brain MRI also indicated no local growth of the lesion where the mass lesion was left.On follow-up after 3 months of steroid therapy, the patient displayed a good response on the imaging studies. MRI revealed that the preexisting mass lesion had regressed extremely. The enhanced small nodular shadow in the lower part of the fourth ventricle had disappeared. Only around the right cavernous sinus, did the findings show a residual small mass of iso signal intensity on T1-weighted images and an extremely low signal intensity on T2-weighted images with no enhancement effect after contrast medium administration Fig. .Fig. 5T2When the prednisolone reaches 25 mg/day, additional treatment using methotrexate in combination with prednisolone is planned for the patient.18F-FDG PET/CT is a valuable tool for the diagnostic evaluation of patients suspected of sarcoidosis and is also important for other major diagnostic discrimination, including lymphoma and metastasis [Intracranial pseudotumors are uncommon, but it is important to recognize such lesions. They may occur in inflammatory diseases , infectious diseases, vascular diseases, and radionecrosis . Among stastasis .Although imaging studies are nonspecific, MRI is a very sensitive diagnostic tool for the finding and detection of intracranial lesions due to neurosarcoidosis . PreviouOne of the typical images of neurosarcoidosis is leptomeningeal involvement which usually appears as a thickening and diffuse and/or focal enhancement , 4, 10. It has been reported that cranial neuropathies, which may involve one or more nerves and occur unilateral or bilaterally , are seeSeveral studies have indicated that increased serum ACE levels may be observed in sarcoidosis, but not in all cases . FurtherAlthough criteria for the pediatric diagnosis of neurosarcoidosis have not yet been established, the most widely used criteria are those developed for adults by Zajicek et al. , 15. DefThe most appropriate treatment for pediatric neurosarcoidosis is remains uncertain. There is no evidence of clinical benefit from surgical removal for neurosarcoidosis , and the


Brown tumor is a rare nonneoplastic focal giant cell lesion that occurs in hyperparathyroidism patients with a prevalence rate of 0.1% in jaws. We report an extremely rare case of brown tumor in mandible of a 40-year-old female patient that presented as the first clinical manifestation of hyperparathyroidism. Dentist played a pivotal role in the present case by the early diagnosis of lesion and its intervention. Hyperparathyroidism (HPT) is an endocrine disorder occurring due to increased secretion of paratharmone resulting in a complex of clinical, anatomical, and biochemical alterations . HPT is One of the skeletal lesions observed in HPT is brown tumor , also teBrown tumor is mostly asymptomatic, but occasionally it may present as a painful exophytic mass . RadiogrWe report a rare case of brown tumor occurring in mandible of a 40-year-old female patient that was the first clinical manifestation and presented as a multilocular radiolucency, which on further biochemical assessment confirmed the diagnosis of adenoma of parathyroid. Along with this case report other giant cell mimickers of oral cavity are also discussed.A 40-year-old female reported to the outpatient department with a chief complaint of pain in the lower left back tooth region since 6 months and associated swelling since 3 months. The swelling was initially small in size and gradually attained present size. Patient gave a history of weight loss since 1 year and traumatic incident 3 months before. Patient was hypertensive since 3 months and is under medication.Extraorally, a swelling was observed on the left lower third of the face Figures  and on iOPG revealed a multilocular radiolucent lesion with well-defined margins was seen in relation to 35 and 36 with thinning out of inferior border of mandible. Loss of lamina dura in relation to 35 and 36 along with loss of continuity of mandibular canal was also observed .FNAC revealed a reddish colored aspirate , composeOn microscopic examination, numerous osteoclast like multinucleated giant cells of varying sizes and shapes which were composed of 10–20 nuclei and dispersed in the background of mononuclear spindle shaped stromal cells were seen. Areas of osteoid, trabecular bone, hemorrhage, and inflammatory component were seen Figures . A giantHematological investigations demonstrated elevated serum calcium and phosphorus levels   along with increased levels of paratharmone . Ultrasound of neck revealed a well-defined hypoechoic lesion of 2.2 × 2 × 3.1 cm, located posteriorly and inferiorly to the right lobe of thyroid causing an indentation which was suggestive of a parathyroid adenoma. Skull radiographs revealed multiple well-defined osteolytic radiolucent lesions in the parietal and occipital areas .Based on the clinical, radiographic, histological, and biochemical analyses, a final diagnosis of brown tumor associated with primary hyperparathyroidism was derived.Primary hyperparathyroidism is the 3rd most common endocrine disease , caused HPT is commonly asymptomatic; however some patients may present with nonspecific symptoms like weight loss, GIT, and musculoskeletal disturbances  which waClassic skeletal lesions like bone resorption, bone cysts, brown tumors, and generalized osteopenia occur in less than 5% of all HPT cases . The incBrown tumor accounts for 10% of all skeletal lesions with a 0.1% incidence in jaws . It is mBrown tumor may involve any part of skeleton but is commonly seen in ribs, clavicle, and pelvis. In head and neck region, mandible is commonly involved compared to maxilla especially the posterior region . The preSymptoms caused by the lesion depend on their size and location. Clinically, brown tumor may present as small asymptomatic swelling in jaws or as a painful exophytic mass which was observed in the present case.Radiographically brown tumors appear as a well-defined unilocular or multilocular radiolucent lesion with expansion of affected bone. Additional features include subperiosteal resorption of phalanges of index and middle fingers, generalized osteopenia, and focal areas of skull demineralization-salt and pepper appearance . SimilarHistopathologically brown tumor exhibits dense fibroblastic stroma, areas of cystic degeneration, osteoid, hemorrhage, macrophages with hemosiderin, and multinucleated osteoclastic giant cells . Cystic Histologically brown tumor mimics many other giant cell lesions of head and neck region. Clinical, radiographic, and histological features of giant cell mimickers are discussed in On biochemical investigations, the present case showed hypercalcemia and hyperphosphatemia, along with increased parathyroid hormone level which aided in the confirmatory diagnosis. These alterations may be due to elevated parathyroid hormone which activates the osteolytic pump causing loss of calcium from bone to extracellular fluid resulting in elevated serum calcium levels. Ultrasound, CT scan, or technetium scan techniques can also be used to detect the diseased parathyroid gland . UltrasoTreatment of HPT is the first step in the management of brown tumor. After parathyroid excision, if the jaw lesions are smaller in size, they tend to regress spontaneously, either completely or partially. If the lesion is large and disfiguring or if the affected bone is weakened, surgical excision of the brown tumors is indicated. Some suggest systemic corticosteroids initially to decrease the size, followed by surgical excision of the residual lesion . RecurreEven though the advancement of various diagnostic process and biochemical tests aids in early diagnosis of HPT, dentists should be aware of possible occurrence of brown tumor involving the jaws of undiagnosed patients as it may be presenting as the first manifestation. Hence it is essential that dentist should have the knowledge about oral manifestations associated with various systemic diseases leading to their early diagnosis.Although the diagnosis of asymptomatic primary hyperparathyroidism is indicated by detection of elevated levels of calcium on routine biochemical analysis, still there is a possibility of patients presenting with advanced bony lesions. Therefore all giant cell lesions occurring in the jaws have to be further evaluated biochemically to rule out primary hyperparathyroidism.


Left ventricular free wall rupture (LVFWR) is a grave complication of acute myocardial infarction (MI). We report a case of a 73-year-old male who developed LVFWR five days after a transmural MI. The diagnosis was confirmed with echocardiography, which showed a large pericardial effusion with a clot in the pericardial sac. This case emphasizes that a high index of clinical suspicion for the acute mechanical complications of MI should be present when managing patients with transmural MIs. In addition, stat echocardiography is necessary to diagnose LVFWR and initiate treatment. Myocardial infarction (MI) is complicated by left ventricular free wall rupture (LVFWR) and pericardial tamponade in 2%-4% of the cases . The freA 73-year-old male presented to the emergency department with a sharp, 7/10, nonradiating, midsternal chest pain for four hours. His past medical history included hypertension, hyperlipidemia, and stage III A non-small cell lung carcinoma, in remission for six years after radiation and chemotherapy. On examination, the vitals were stable. The cardiovascular examination was normal and the respiratory exam indicated wheezing. The chest X-ray showed pneumonia in the left lung base. A 12-lead electrocardiogram showed ST segment elevation in leads III and aVF consistent with an acute inferior wall MI  and was started on norepinephrine for hypotension. The electrocardiogram showed no new ST segment changes. On examination, he was diaphoretic and in no respiratory distress. Jugular venous pressure was mildly elevated and heart sounds were not audible. A stat echocardiogram showed a new large pericardial effusion with a clot in the pericardial sac. There was a muscular dehiscence in the left ventricular apex, indicating a likely site of myocardial rupture Figure . The lefAn MI complicated by LVFWR carries a high mortality rate. The underlying risk factors causing free wall rupture are: anterior location of the infarct, large transmural infarct, age > 70 years, female sex, no history of previous angina or MI, peak CK-MB above 150 IU/L, ST-segment elevation or Q-wave development on the initial electrocardiogram, and late or failed PCI -4.In a large transmural MI patient without a history of angina or prior MI, there is an increased risk of rupture because there is no collateral circulation present, which would help minimize the development of necrotic tissue . AdditioLVFWR is clinically divided into two subtypes, acute and subacute. In the acute form, there is a cardiac arrest due to severe hypotension and electromechanical dissociation secondary to pericardial tamponade. The subacute form presents with persistent or recurrent chest pain, nausea, restlessness, electrocardiogram changes of localized or regional pericarditis, moderate to severe pericardial effusion, sinus bradycardia or nodal arrhythmia, and sudden hypotension or death ,5.Echocardiography is crucial for the prompt diagnosis of LVFWR. The finding of pericardial effusion on echocardiography is 100% sensitive, but has low specificity, as the other causes of pericardial effusion after MI are pericarditis, hemorrhage from postinfarcted myocardium, and coronary perforation. Moreover, echocardiography can also show an echogenic layering in the pericardium due to a pericardial thrombus, regional myocardial dilation, and an abnormally thin and akinetic myocardium. In color-flow Doppler echocardiographic evaluation, a color-flow jet visible through a wall defect is diagnostic of LVFWR. However, if a pericardial thrombus covers the rupture site, a color-flow jet would be absent. If the patient is stable, then cardiac computed tomography (CT) and magnetic resonance imaging (MRI) could be considered .Once the diagnosis of myocardial rupture is made, management starts with aggressive volume resuscitation with inotropic and vasopressor support and percutaneous circulatory support, such as intra-aortic balloon counterpulsation and extracorporeal membrane oxygenation. Furthermore, the patient can undergo a surgical repair of the rupture, which includes an infarctectomy or patching. The outcome of surgery depends upon the degree of hemodynamic instability, the location of the rupture, and the weakened state of the surrounding infarcted myocardium. A weakened necrotic surrounding myocardium is a weak anchoring site for sutures in surgical repair. Therefore, when an infarctectomy is performed, it extends to include the viable myocardium, thereby decreasing the size of the left ventricular cavity. In contrast, a Dacron or Teflon patch could be used. These patches are applied with cyanoacrylate glue and cover the area of rupture, the surrounding necrotic tissue, and part of the viable myocardium. Hence, they do not decrease the size of the left ventricular cavity . AdditioIn patients with transmural MI, total coronary occlusion is an essential element in causing a myocardial rupture . In turnOur patient had a transmural infarction, but after the revascularization of the lesion, the TIMI flow was low. Therefore, he had the mechanical complication of LVFWR with a cardiac arrest. We were able to diagnose this fatal complication, but no intervention was undertaken, as the prognosis was poor. Therefore, through this case, we would like to suggest that LVFWR is an important differential diagnosis to consider in unstable patients who have had a recent transmural MI in order to have early intervention and improve mortality outcomes.


Ovarian sarcomas represent less than 1% of all ovary cancers and usually are frequent in adults. Primary angiosarcomas are exceptional in the ovaries within children.We reported a case of primary ovarian angiosarcoma in a 12-year-old girl in a resource-constrained context. Immunohistochemistry study showed the positivity of CD34, CD31, factor VIII, while S100 was negative. The diagnosis of primary non-metastatic angiosarcoma was retained. She was unable to undergo the CWS-2002P chemotherapy since her parents could not afford it.This case report described a rare type of a primary ovarian angiosarcoma within a child, diagnosed in a low-income country in a laboratory with limited resources. Ovarian sarcomas are very rare and represent less than 1% of all malignant ovarian tumors , 3. AS aA 12-year-old female had consulted at the Department of Gynecology for pelvic pain for 22 days which was intense in the evening at lying. She had a polyuria for 10 days and occurred over the night where she woke up 7 times on average to urinate. There was no notion of traumatism. There was no medical and surgical history. There was no concept of sexually transmitted infections. The socio-economic level was considered average; she had never smoked and did not drink alcohol. She had normal pubertal development with chest changes and pubic hair.2. The blood pressure was 110/60mmhg. The general condition was good. The patient suffered from pain in the right pelvic region triggered by palpation, and thus, limiting the movement of the right hip. Palpable adenopathy was absent. The remaining physical examination was normal. The biological examination was normal. The abdominal ultrasound showed a solid mass that occupied the right ovary. The biological check-up was in normal. Alpha-fetoprotein and β-hCG were normal. Ultrasound of the abdomen showed a solid mass that occupied the right ovary.Clinical examination displayed a temperature of temperature at 37 °C, a weight of 46 kg for a size of 1.34 m, BMI was 25,62Kg/mThe patient underwent an ovariectomy. The macroscopic analysis revealed that the ovariectomy was grayish, irregular and elastic and measured 17cmx14cmx9cm. The ovarian sections were heterogeneous, including red areas and hemorrhagic territories. The normal ovarian tissue was not identified. The Fallopian tube was normal. The contralateral adnexa and the uterine body were normal. The peritoneum was apparently normal and the peritoneal cavity contained approximately 1000 mL of blood ascetic fluid.The sections of the ovarian tumor were made and ten blocks of paraffin were made. Conventional histology showed a malignant vascular tumor proliferation made of many very small vascular luminesces with endothelial cells exhibiting nuclear atypia and mitoses. The necrosis was present. As diagnostic hypotheses, we primarily reported angiosarcoma followed by hemangiopericytoma Fig. . Three bThe result of immunohistochemistry analysis was CD31+, CD34+, and Factor VIII+ Figs. , 3 and 4Angiosarcomas are uncommon vascular cancers, accounting for 1–2% of soft tissue sarcomas with high aggressive and metastatic potential . AngiosaAngiosarcoma has been identified as the second most frequent sarcoma in germ cell tumors with sarcomatous components after rhabdomyosarcoma . This raThe histopathological diagnosis of angiosarcoma is challenging. Angiosarcoma may be confused with vascular tumors of intermediate malignancy  , 3, 4. HThese abnormal endothelial cells typically form vascular sinusoids that infiltrate the surrounding tissue. The vessels may be lined by a single or multiple layers of malignant cells, and thus creating an irregular network of vascular lumina. The vascular spaces may be difficult to recognize due to the solid appearance of the tumor in case of poorly differentiated angiosarcoma . MitoticImmunohistochemistry (IHC) is relevant for the certainty of angiosarcoma diagnosis. This technique carried out in a Laboratory of Anatomic Pathologyin France revealed the positivity of specific markers of the angiosarcoma such as the factor 8, CD31, and CD34. It is clearly shown that IHC is an essential technique in the diagnosis of undifferentiated angiosarcomas . CD34 maAngiosarcomas badly respond to chemotherapy and radiotherapy. Surgery is the key treatment of the early form of this cancer although there is common recurrence . An optiThe prognosis of angiosarcoma in general is dismal, with a 5-year survival rate lower than 30% for non-metastatic angiosarcoma, and an over-all survival of 8 months for the metastatic stage . AlthougAngiosarcomas are extremely rare vascular tumors in children and ovarian localization is exceptional with high mortality. Its diagnosis must be sought in tumors with a rich vascular network, necrosis and significant mitotic activity. This observation also shows the diagnostic difficulties encountered by pathology laboratories under equipped in developing countries and poses the problem of therapeutic management in our country.


Extramedullary tumors composed of myeloblasts or monoblasts can present in various locations. Patients with a history of acute myeloid leukemia (AML) can present with neuropathic pain and no evidence of relapse of their leukemia. Neuroleukemiosis is a form of extramedullary tumor present in the peripheral nervous systems (PNS) of leukemia patients. We report two AML patients who were in remission and later presented with neurological symptoms due to neuroleukemiosis with negative bone marrow biopsies. Acute myeloid leukemia (AML) is a malignant disease of hematopoietic stem and progenitor cells . It is cThe literature contains only a handful of case reports of isolated leukemic masses infiltrating the PNS. Such masses usually appear after patients are in remission and there are no other signs of relapse . We repoCase report 1An 18-year-old male with a history of AML diagnosed in 2010, status post bone marrow transplant and donor lymphocyte infusion (DLI), presented with a one-year history of progressive deep left shoulder and left upper arm pain extending into the C8 dermatome. Epidural injection did not relieve his pain and in a follow-up exam, he had contralateral symptoms and right S1 radicular symptoms. Analysis of the cerebrospinal fluid (CSF) was positive for leukemic cells. Initial magnetic resonance imaging (MRI) showed only C7 nerve involvement on the left. Repeat MRI showed multiple expansive soft tissue masses extending through the neural foramina involving the right C4-C5 and C5-C6, the left neural foramina at C6-T3 into the paraspinal tissues, and the right L5-S1 neural foramina  and PNS involvement is rare -7. Both The spread of leukemia to the peripheral nervous system is still debated. A blood–nerve barrier (BNB), similar to the blood–brain barrier (BBB), separates the circulating blood from the peripheral nerves -7. The BWang, et al. suggested that in patients with isolated neuroleukemiosis, a hematogenous spread of blast cells into the PNS is more likely than longitudinal dissemination from nerve root . They prNon-resolving neuropathic symptoms of pain, numbness, and weakness can be the first signs of neuroleukemiosis. In the cases reported here, both the patients presented with some type of neuropathic symptoms. In the first case, the patient presented with progressive shoulder and arm pain over a year. The symptoms of left arm weakness, however, seemed to progress much more rapidly in the second case than the first. With only neuropathic symptoms present, it is easy to misdiagnose a leukemic mass infiltrating the PNS, especially if the blood smear and bone marrow biopsy are negative for leukemia, which was not the case with these patients.Differential diagnosis for neuroleukemiosis includes Guillain-Barre syndrome (GBS), neurotoxicity from chemotherapy, and abscess ,6. NerveTreatment decisions depend on the type of leukemia, patient symptoms, and extent of metastasis. Local infiltration and local tumors are most frequently treated with local radiation therapy and surgical decompression to alleviate symptoms ,10. MostIn patients with unexplained new-onset neuropathy and a history of AML, neuroleukemiosis must be included in the differential diagnosis. Even when there is apparent bone marrow remission and blood smears are negative, the possibility of CNS or PNS involvement by relapsing leukemia cannot be ruled out. Because it is difficult for systemic chemotherapeutic drugs to penetrate the BNB, the PNS makes an ideal location for blast cells to infiltrate and thrive after the patient has been in remission for many years.


In extremely preterm infants, acute abdominal emergencies are fortunately less common with improving care. Spontaneous intestinal perforation and necrotizing enterocolitis are conditions where emergency surgery is most often needed. Conservative medical management and placement of temporary drain are often used in the initial management. Internal hernia (IH) is an uncommon cause of bowel obstruction in neonates, is difficult to diagnose and unfortunately are found only at autopsy. The presentation in preterm infants, distinction between these conditions, and the need for early diagnosis of IH are discussed. Internal hernia (IH) is an intra-abdominal herniation of viscera through a normal or abnormal aperture within the peritoneal cavity . IH is aA 35-year-old woman with no previous medical history with regular prenatal care and normal screening tests was admitted at 23 weeks’ gestation due to premature rupture of membranes. She received betamethasone, antibiotics, and magnesium but 5 days later she went into labor and underwent emergency caesarian section for fetal bradycardia and suspected cord prolapse. A female infant was delivered at 24 weeks’ gestation with a birth weight of 625 g and required mechanical ventilation for 3 days and subsequently non-invasive ventilation. Trophic feeds and parenteral nutrition were initiated within 24 h of life. At 17 days of age, when she was tolerating close to her goal volume of feeds, she started having increasing abdominal distension and feed residuals (aspirates). Serial abdominal radiographs showed bowel dilation, which appeared fixed without evidence of pneumatosis or free intraperitoneal air  and necrotizing enterocolitis (NEC) are by far the most common , 6. IH iThe initial clinical course of the infant described above, with feed intolerance, abdominal distension, and dilated bowel loops could be attributed to mechanical obstruction especially in the first week of life and the differential diagnosis could include atresias, sepsis-related ileus, meconium plug or ileus or SIP. Events like apnea and bradycardia, hemodynamic instability with metabolic acidosis, disseminated intravascular coagulopathy with tense, firm, and discolored abdomen is usually assumed to worsening NEC or sepsis, especially after the first couple of weeks of age, when infants have established feeds. Initially, bowel rest, decompression, and broad-spectrum antibiotics are often instituted. Surgical consultation and intervention are more frequently done in the former group with early signs of obstruction and in the latter group is generally reserved for the patients with pneumoperitoneum and other signs of severe NEC or if there is a definite evidence of mechanical obstruction. The surgical management of a critically ill extremely preterm infant with diagnosis of severe NEC would be placement of a peritoneal (Penrose) drain, as there is a general reluctance to embark on a laparotomy in unstable infants. Systematic reviews, analyzing randomized controlled trials have not been able to answer, whether peritoneal drainage or laparotomy as treatment for perforated NEC, affects mortality or long-term sequelae or neuro-developmental outcomes , 7. NeitSonography is being used as a complementary diagnostic modality to abdominal radiographs in infants with NEC, SIP, and malrotation –10. MultHirata has previously described two preterm infants who both died due IH and the authors lamented that “the delay in diagnosis of organic intestinal obstruction leads to fatal outcome in VLBW (preterm) infants” . PretermIn extreme low-birth weight infants with signs of feed intolerance, abdominal distension, or those with signs of NEC, US may be a useful modality for initial and sequential assessment of bowel health. In those with worsening clinical signs (as in suspected NEC in the absence of pneumatosis), it may be reasonable to embark on an exploratory laparotomy rather than continued conservative management or peritoneal drainage, given that there have been marked improvements in perioperative care, especially as conditions like IH or malrotation could be missed.This single case study with no identifying details was given a waiver by the institutional review and ethics board.RK designed study, collected data and wrote the manuscript.The author declares that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.


Gastrointestinal bezoars, collections of incompletely digested material within the alimentary tract, can present as a diagnostic challenge and should be considered in the differential diagnosis and management of small bowel obstruction, ischemic bowel, or bowel perforation. We present a case of a 37-year-old man with a distant history of laparotomy for superior mesenteric artery thrombosis requiring partial small bowel resection of the jejunum who presented with worsening abdominal pain, nausea, vomiting, and hematemesis. An abdominal computed tomography revealed dilated loops of small bowel with a transition point at the ileum, distal to his prior bowel anastomosis. He was managed initially nonoperatively, but persistent vomiting and worsening distention necessitated urgent exploratory laparotomy. During the procedure, a 4 cm by 3 cm phytobezoar was discovered at the midjejunum. The patient had an unremarkable postoperative course with no further symptoms at 1-year follow-up. Timely diagnosis and treatment of bezoar is essential to minimize patient complications. Gastrointestinal bezoars are rare but often can present with serious complications if untreated. While the physical nature of these bezoars varies, the extent to which they can affect the motility of the alimentary tract is also variable, creating a challenge both in diagnosis and management. In this case, we present the clinical, radiographic, and operative findings of a man who presented with a phytobezoar in the setting of a small bowel obstruction. We also review the existing literature to address the appropriate diagnostic and treatment strategies to minimize perioperative complications.The patient is a 37-year-old man with a past medical history of hypertension, diabetes mellitus, extensive hypercoagulable history, and a remote history of exploratory laparotomy for an embolectomy of the superior mesenteric artery and a small bowel resection of the jejunum. He presented to the Emergency Department with an 8-hour history of worsening, crampy abdominal pain, nausea, vomiting, and multiple episodes of hematemesis. He had a bowel movement prior to arrival, but otherwise his review of systems was unremarkable. Physical examination was notable for hemodynamic stability and a mildly distended abdomen with epigastric tenderness but without guarding or rebound tenderness. Given his prior surgical history, a computed tomography (CT) of the abdomen was obtained, which showed dilated loops of bowel with a transition point in the region of the proximal ileum, just distal to his prior bowel anastomosis. The distended proximal small bowel loops contained fecalized material, and the distal small bowel loops were also decompressed without evidence of ascites, bowel wall enhancement, or free air .The patient was admitted and initially managed nonoperatively with fluid resuscitation, pain control, bowel rest with nasogastric tube decompression, and serial abdominal exams. Following return of bowel function, the nasogastric tube was discontinued on hospital day 2 but reinserted hours later due to persistent, new-onset vomiting, and worsening abdominal distention. Without significant improvement in symptoms, the patient was taken to the operating room urgently for an exploratory laparotomy. Intraoperatively, the small bowel was dilated to approximately the midjejunum, where an intraluminal obstruction was palpated and subsequently identified to be a phytobezoar. Consequently, a longitudinal enterotomy was performed to evacuate the phytobezoar measuring 4 cm by 3 cm and was closed in a transverse fashion in two layers . The phyGastrointestinal bezoars are collections of incompletely digested material within the alimentary tract that can lead to serious medical complications including small bowel obstruction, bowel ischemia, or perforation. Bezoars are classified by their contents: phytobezoars involve fibrous nondigestible matter from fruit, vegetables, psyllium, or persimmon (diospyrobezoar); trichobezoars comprised ingested hair, prevalent in women aged less than 30 years; pharmacobezoars arise from enteric-coated aspirin, extended release capsules (nifedipine and theophylline), or sucralfate; and occasionally, bezoars result from ingested styrofoam, fungi, cement, or other inedible objects. While phytobezoars are the most common types of bezoar, phytobezoar-induced small bowel obstruction is a rare clinical situation, accounting for less than 5% of all mechanical intestinal obstructions .Patients with a history of peptic ulcer disease, previous gastric surgeries or bowel resections, and systemic diseases that reduce gastrointestinal motility or cause strictures  are predisposed to bezoar formation . ExcessiIn patients with small bowel obstruction due to phytobezoar, the ileum is the most common site of obstruction . Two recRadiographically, phytobezoars are well-circumscribed intraluminal masses that have a heterogeneous appearance, often with a mottled, air-bubble pattern and an absence of mesenteric fat stranding . They caDiagnosis of bezoar is often delayed, but once established, there are several treatment options available. Medically, treatment with prokinetic agents such as itopride, mosapride, and metoclopramide or enzymatic breakdown (dissolution) is often sufficient. If the lesion is confined to the stomach or duodenum, endoscopic fragmentation with forceps or snares can be employed to diagnose and deconstruct the bezoar with a goal of alleviating the bowel obstruction. Additionally, for gastric bezoars, Coca-Cola has been shown to be an effective dissolution agent . For thoTimely diagnosis and treatment of bezoar is essential to minimize patient complications, although in most scenarios, this task is difficult to accomplish. In the current era, the utility and dependence of imaging to aid in the diagnosis cannot be underestimated. Therefore, bezoar obstruction, although rare, should be an important consideration, especially during the initial evaluation and workup of patients presenting with abdominal symptoms to the emergency room.


Rapidly involuting congenital hemangioma is a congenital soft tissue tumor, which is difficult to excise completely and rather prone to recur. This atypical tumor combined with capillary-lymphatic-venous malformation was not reported in the literature.We report an atypical case of a 16-year-old teenager who was born with a mass in his right upper limb. Since there is a recurrence after excision for several times and had a serious impact on daily life, we chose amputation. After the surgery, the patient gained a functional recovery. Two years after the surgery, he had no tumor recurrence.For this rare tumor with repeated recurrences and significant impact on daily life, we suggest performing amputation at the early stage. Rapidly involuting congenital hemangioma (RICH) is an uncommon, often high-flow vascular tumor that presents at birth. It is generally considered as a congenital condition . BecauseA 16-year-old teenager who was born with a mass in his right hand, which was diagnosed as hemangioma. When he was 5 months old, the tumor was partially excised in Shanghai. The mass developed increasingly after the surgery and presented diffuse growth from the distal to proximal upper limb. The patient was treated with sclerotherapy at the age of three in Zhengzhou but the treatment failed. He was treated with excision again and abdominal pedicle flap surgery at the age of seven in Beijing. One year later, his fingers were separated. And the tumor had been partially excised for several times in Wenzhou before the patient came to our hospital. The patient’s right upper limb could not move upward. He was almost helpless in his daily life and not able to go to school. The physical examination showed multiple scars on his right upper limb and a huge mass in his right hand Fig. . Gross eIn this case, we had difficulty in deciding whether the tumor should be one type within the ISSVA classification. Infantile hemangioma is the commonest tumor in infants with progressive proliferation in the first year of life and slow regression in the next few years. There are also few other rare congenital hemangiomas  with different clinical courses –8. CliniTreatment choice is difficult for this rare case. En bloc resection of the tumor may be the best treatment to prevent recurrence. But due to the nerves, tendon, ligament, and essential structures surrounding the tumor, it will be easy to cause the physical disability if the tumor is excised completely. Patient in this case involved all tissues and growth is progressive after partial resections. The main purpose of the surgery is to release the local symptoms such as persistent pain and numbness, increasing size of the mass, and functional impairment. The patient in this case had several aggressive surgeries, which not only stimulated the growth of the tumor but also resulted in unnecessary injuries in other parts such as the donor site. Therefore, it needs to avoid similar crisis in the future. A prosthesis was fitted after the amputation, the patient could raise his shoulder, and his ability of self-care was obviously improved. Sclerotherapy, chemoembolization, and embolotherapy  are treaFor this rare tumor with repeated recurrences and significant impact on the daily life, we suggest performing an amputation at the early stage.


Goldenhar´s syndrome (GS) also known as oculo-auriculo-vertebral spectrum (OAVS) is a relatively rare condition. GS is of multifactorial etiology that includes environmental and/or genetic factors, in addition to teratogens that disturb the blastogenesis. A 5-year-old girl from Saudi Arabia, was a member of dizygotic twins conceived by assisted reproductive technology (ART), and born with features of GS. She had asymmetrical face, cleft lip and palate, right microphthalmia and microtia. Radiological imaging showed right maxillary and mandibular bone hypoplasia, asymmetrically enlarged parietal foramina, a persistent midline occipital foramen, abnormal bone arising from occipital bone, extending along tentorium cerebelli, and a lipoma at the right tentorium cerebelli. A rudimentary right eye with dermoid cyst and pseudotumor as well as bilateral atresia of external auditory canals were present. Karyotyping was normal. ART and the risk of manipulation of ovum/embryo, was shown to be associated with multiple gestation and an increased risk of major birth defects. Given that our patient was from Eastern-province close to the South of Iraq, where Gulf wars took place and the reported incidence of birth defects, including orofacial malformation, jumped there to about seven-folds, after war, thus, environmental contamination, and the possible teratogenic effect of depleted uranium could not be excluded. In conclusion, our case of GS, disclosed a rare radiological finding in calvarial anatomy, and asserted that, careful clinical evaluation is recommended in cases of GS. ART fertilization risk along with the possible parental environmental exposure were regarded as the potential cooperators of multifactorial etiology in our case. Goldenhar´s syndrome (GS) also known as oculo-auriculo-vertebral spectrum (OAVS) is a relatively rare congenital condition characterized by incomplete development of the head and face structures. GS was first described in 1845 by von Arnt, then in 1952 Maurice Goldenhar reported its characteristic features. In 1963, Gorlin named this syndrome as OAVS. A wide range of anomalies were described in OAVS including unilateral or bilateral facial hypoplasia, ear, and eye malformations. In addition, vertebral, cardiac, renal, and cerebral abnormalities were reported to be part of the spectrum. Ear malformation as a minimal clinical sign, must be present, to raise the index of OVAS suspicion. GS occurs in about 1 in 3500 to 1:5600 live births. Although, the exact cause still unknown, a multifactorial etiology that included interaction of environmental and/or genetic factors, in addition to teratogens, were proposed to disturb the blastogenesis. Some of the reported cases had a positive family history (2%), suggestive of an autosomal dominant/ recessive inheritance [GS represents anomalies of structures arise from the embryonic first and second branchial arches, the first pharyngeal pouch, the first branchial cleft, and the primordia of the temporal bone. GS could be resulted from failure of the embryonic blood supply during the critical period from third to the fifth week in the development of the craniofacial structures. Although this circulatory failure occurs before the presence of ossification centers in the mandible and maxilla, it could cause abnormal bone formation . PoswillThe purpose of this paper is to report rare cranial anomalies among the growing list of associated and underestimated abnormalities of GS, and to emphasize the multifactorial nature of this syndrome in our case.A 5-year-old girl from Saudi Arabia who was diagnosed as a case of GS, referred on March 2016, to the radiology department in Ibn Al-Nafees Hospital in Manama- Kingdom of Bahrain, by a maxillofacial surgeon for a computed tomography (CT) scan of the head and neck. Our patient is the only live birth child of a young non-related couple, who tried to conceive again after 3 years following a spontaneous first-trimester miscarriage, and an ectopic pregnancy, using medications. Given the 28-year old mother had polycystic ovary and a troublesome medical history, an intracytoplasmic sperm injection (ICSI) procedure was done and (3 embryos were transferred). Our case was the living member of a dizygotic twins whilst the other member of the twins was miscarried at the 11th week of gestation. The mother had a long history of hypochromic microcytic anemia with a hemoglobin range of (7-9 gm/dl), and supposed to be a carrier of alpha thalassemia. During the 12th gestational week of our case, the mother had progressive pallor and her hemoglobin dropped down to (6 gm/dl), necessitated blood transfusion. Then at the 26th week, the mother had signs of preterm labor, which was halted using medication, and the pregnancy was ended by Cesarean section. Of note, the mother who was allergic to a long list of drugs, received hormonal therapy to trigger or regulate ovulation, drugs before and after performing ICSI, then during pregnancy, she received folic acid 5mg/day, and calcium was started from the 4th month of pregnancy. Soon after delivery, the mother who had 5 years history of migraine and tingling sensation in her limbs, developed an epileptic attack and proved to have multiple sclerosis. There was no gestational diabetes, no history of smoking, and no family history of craniofacial malformations. According to the declaration of Helsinki, written informed consent was obtained from the parents of the patient for publication of this Case report and the accompanying images.After birth, the full term baby who was diagnosed antenatally to have cleft lip and palate, was found to have dysmorphic features and intrauterine growth restriction. Due to upper airway obstruction related to her craniofacial malformation, mechanical ventilation and tracheostomy tube were used for a while. After being stabilized, she was referred to ENTist for repairing cleft lip at 6 months of age, and cleft palate and preauricular ear tags at 18 months. Then she was referred to ophthalmologist, pediatric and plastic surgeons, and maxillofacial specialist for intervention. As shown in Figure Our case has the characteristic features of GS with the additional presence of calvarial bone defects. Although skeletal defects were reported in most cases of GS, calvarial bone defects and parietal bone in particular, was rarely described. Given the accessible published literatures since the 1960s, we could find 2 cases of parietal bone defects, whereas enlarged parietal foramina and a persistent occipital foramen were not reported (Table Assisted reproductive technology (ART), such as ICSI and the risk of manipulation of ovum and embryo, was shown in general to be associated with multiple gestation and an increased risk reaching up to 10% for major birth defects in some studies compared to naturally conceived embryos . Our casOur patient is of Saudi nationality, living in the Eastern province, Al-Qateef city, which borders the Arabian- Persian Gulf, and it is the place for most of Saudi Arabia's oil production and global hub for chemical industries. The Eastern province had reported the second highest frequency of birth defects, including GS next to the capital Al-Riyadh, together they accounted for about half of cases of birth defects in the country . Taken tTo the best of our knowledge this is the first report of GS presenting with bilateral irregularly and asymmetrically enlarged parietal foramina and a persistent occipital foramen in addition to the presence of the abnormal bone arising from the occipital bone. Accordingly, careful imaging and clinical evaluation are recommended in cases of GS. ART fertilization risk along with the possible parental environmental DU exposure risk, were regarded as the top potential cooperators of multifactorial etiology in our case.


Glial choristoma is considered to be a type of brain heterotopia consisting of ectopic central nervous tissue. We herein report a neonate with glial choristoma of the tongue who developed respiratory distress due to airway obstruction. A male neonate presented with respiratory distress due to a soft mass on the midline region of the dorsal tongue base at birth. He was intubated using a flexible fiberoptic nasopharyngoscope. MRI showed a well-circumscribed mass measuring 25 × 23 × 27 mm in size in the same region. A histologic examination confirmed a pathological diagnosis of glial choristoma. He underwent tracheotomy at 22 days of age, and a subtotal resection of the tumor was performed at five months of age. The clinical behavior of oral glial choristoma varies depending on the age at onset as well as the location and size of the mass. The small size of the organ and the narrow operating field hamper the surgical approach in neonates. The optimal therapeutic strategy for neonatal cases of glial choristoma should thus be determined based on the condition of each individual patient. Choristoma generally consists of heterotopic tissue that is microscopically normal. Glial choristoma is considered to be a type of brain heterotopia that comprises ectopic central nervous tissue. It usually presents in the head and neck region. The nasal area is most often affected , and theWe herein report a neonate who presented with glial choristoma of the tongue who developed respiratory distress due to airway obstruction.A male neonate was born by normal vaginal delivery at 37 weeks of gestation. His birth weight was 3010 g (72th percentile), height 48 cm (58th percentile), and head circumference 35 cm (95th percentile). His 29-year-old mother had gestational diabetes mellitus controlled by the dietary treatment. Polyhydramnios had been detected by ultrasonography at 33 weeks of gestation. Neither fetal ultrasonography nor magnetic resonance imaging (MRI) revealed any abnormal findings accounting for polyhydramnios.He presented with symptoms of respiratory distress associated with tachypnea, cyanosis, and retractive breathing at birth. Even when the baby attempted to cry, his voice could barely be heard. An immediate observation of his oral cavity identified a soft mass on the midline region of the dorsal tongue base. Due to progressive respiratory distress with marked hypercapnia, he was intubated using a flexible fiberoptic nasopharyngoscope, because the mass was prone to bleeding by contact. Contrast-enhanced computed tomography (CT) imaging revealed a low-density, nonenhancing 20 mm wide mass on the midline region of the tongue base. MRI showed a well-circumscribed mass measuring 25 × 23 × 27 mm in size in the same region with a low signal intensity on T1-weighted images  and a hiA biopsy was performed at 15 days of age. A histological analysis of the biopsy specimens revealed a subepithelial eosinophilic fibrous mass without a capsule. Mature glial tissue characterized by spindle-shaped cells with eosinophilic cytoplasm and slight myxoid change without atypia were observed . The celCurative surgery during the neonatal period was deemed too risky, so he underwent tracheotomy at 22 days of age. Since his difficulty in swallowing had probably been caused by the tongue mass, he was fed through a nasogastric tube. His respiratory condition remained stable except for short-term hospitalization due to acute bronchitis at four months of age, when the mass on MRI was noted to be almost the same size as that observed at birth.He underwent subtotal resection of the mass at five months of age. The postoperative movement of the tongue and the vocal cords during phonation were good. Although he was able to swallow milk and solid foods, he remained dependent on nasal feeding due to the slight aspiration of liquid, probably resulting from a very mild complication affecting the movement of the epiglottis.Glial choristomas are rare congenital head and neck lesions seen most commonly in neonates or in early childhood. They consist of differentiated neuroectodermal tissue and represent developmental heterotopias rather than true neoplasms . The nasAlthough a surgical resection is the standard treatment for glial choristoma, the optimal timing for surgery remains unclear and probably depends on the condition of each patient. While early resection enables extubation and oral feeding , the smaGlial choristoma should be considered in the differential diagnosis of neonatal airway obstruction. It may be best to determine the therapeutic strategy for neonatal cases of glial choristoma based on the condition of each individual patient.


Clip migration into the common bile duct (CBD) is a rare but well-established phenomenon of laparoscopic biliary surgery. The mechanism and exact incidence of clip migration are both poorly understood. Clip migration into the common bile duct can cause recurrent cholangitis and serve as a nidus for stone formation. We present a case, a 54-year-old woman, of clip-induced cholangitis resulting from surgical clip migration 12 months after laparoscopic cholecystectomy and laparoscopic common bile duct exploration (LC+LCBDE) with primary closure.  Laparoscopic cholecystectomy and laparoscopic common bile duct exploration (LC+LCBDE) is currently a widely use technology for patients with gallstone and choledocholithiasis. Clip migration into the common bile duct (CBD) is a rare complication of laparoscopic biliary surgery. Surgical clip migration into the common bile duct can cause recurrent cholangitis and serve as a nidus for stone formation. Up to date, few cases of surgical clip migration have been reported in the literature. The etiology and exact incidence of clip migration are both unclear. We report a case of Hem-o-lok clips migration 1 year after laparoscopic cholecystectomy and laparoscopic common bile duct exploration (LC+LCBDE) with primary closure. The patient was successfully treated with endoscopic sphincterotomy plus balloon dilation (ESBD) and Hem-o-lok clips extraction. The patient improved uneventfully following the procedure. We hope that this case draws laparoscopic surgeon's attention to this rare phenomenon.A 54-year-old woman had undergone a successful LC+LCBDE in our hospital on December, 2015. The operation had been performed without difficulty and we had used 2 Hem-o-lok for the duct/artery. Subsequently, primary closure of the incision of CBD was performed with an interrupted 5-0 absorbable suture. A bile leakage was detected on postoperative day 2 and spontaneously resolved after 18 days of conservative treatment. The patient was discharged uneventfully on postoperative day 20.On January, 2017, this patient was readmitted for intermittent upper abdominal pain for a month without fever and jaundice. Physical examination revealed tenderness in the right upper quadrant of her abdomen. Laboratory examination showed no abnormal parameters. Abdominal US revealed a mildly dilated biliary tree with no visualized CBD stone. Magnetic resonance cholangiopancreatography (MRCP) showed a slightly dilated common bile duct with a low signal filling-defect in the distal common bile duct, considering CBD stone . During Advances in laparoscopy have made LC+LCBDE a widely accepted strategy for patients with gallstones and choledocholithiasis. The single-stage surgical strategy has been shown to be safe, effective, and cost-effective with shorter hospital stays . ExposurPostoperative clip migration is a rare but well-established complication. Migration into the common bile duct after laparoscopic cholecystectomy was first reported in 1992 . Apart fHowever, the exact mechanism of this condition remains controversial. Multifactor may contribute to the process: the first possible pathogenesis is inappropriate application of clips including incomplete closure of cyst duct and incorrect placement of clips that result in biloma. The number of endoclips used during the initial operation is also an important factor , 8. The To prevent the incidence of clip migration, all the technical factors in the surgery should be considered: confirming the relationship of Calot's triangle during dissection, minimizing the number of clips, and avoiding unnecessary surgical procedures . The plaClinical manifestations of clip migration-induced cholangitis are similar to those of noniatrogenic ones, usually present with abdominal pain, fever, and obstructive jaundice. In our case, pure abdominal pain made us consider the postcholecystectomy syndrome at first. Thus, it emphasizes the importance of early recognition of this complication when patients present with upper abdominal pain or symptoms of biliary obstruction after LC. The diagnosis is suspected on the basis of noninvasive imaging. Ultrasonography is cheap, widely available, and safe and is recommended for patients with suspected foreign body. CT scan is widely used to investigate patients with pain or other abdominal symptoms. MRCP provides clear anatomy of biliary tree with a high sensitivity and specificity.ERCP is a superior approach to manage the complication with a high success rate of about 85% . In our In conclusion, postoperative clip migration has been a well-recognized phenomenon ever since their first use in surgery, albeit rare. Patients who had LC and LCBDE should have a careful surveillance and strict follow-up to ensure the safety. The first investigation might be MRCP and then ERCP for subsequent management.


We present the case of a patient with acute myelomonocytic leukemia with trisomy 8 and novel translocation t. In addition to acute leukemia she had debilitating left shoulder arthritis due to granulocytic sarcoma formation in the joint space. Her shoulder pain did not improve during induction chemotherapy but she experienced rapid relief of symptoms with use of local radiation. Her leukemia was found to be primary refractory to chemotherapy and despite an attempt at salvage therapy she died 2 months after diagnosis. A 60-year-old previously healthy female presented with a 3-week history of increasing left shoulder pain; one week of nausea, abdominal pain, fevers, chills, and new-onset confusion. Laboratory evaluation demonstrated a white blood cell count of 22,600 per cubic millimeter with 28% blasts, hematocrit of 44%, platelet count of 102,000 per cubic millimeter, and calcium of 15.2 mg/dL. AML subtype M4 (according to the French-American-British classification) was confirmed by bone marrow biopsy. Cytogenetics showed trisomy 8 and t. Bone scan and bone survey were unremarkable. The patient was given standard induction therapy with cytarabine and idarubicin. Zolendronic acid was given for hypercalcemia. Left shoulder pain continued to worsen. Day 14 bone marrow biopsy showed persistent blasts; thus salvage therapy with mitroxantrone and etoposide was initiated. Shoulder pain improved partially, MRI of the left shoulder was performed showing a soft tissue mass in the glenohumeral joint space with cortical erosion of the proximal humerus . Biopsy This case presents three unusual findings; granulocytic sarcoma within the joint space, a translocation not previously reported in AML, and hypercalcemia as a presenting sign of acute myelomonocytic leukemia.Granulocytic sarcoma is a collection of granulocytic precursor cells, named chloroma for its greenish appearance under the skin due to presence of myeloperoxidase. It is usually associated with AML, particularly subtypes 4 and 5, or the blastic phase of CML. Granulocytic sarcoma may present anywhere in the body, but most commonly appears in the skin, soft tissue, lymph nodes and bone . When foLeukemic arthritis is typically pauciarticular involving large joints, but may also present as a polyarthritis similar to rheumatoid arthritis , 6. LeukReview of literature shows very few cases of granulocytic sarcoma in the joint space. One case involved the glenohumeral joint in a patient with essential thrombocytosis, and another case involved the hip joint in a patient with acute promyelocytic leukemia , 13. AddOur patient had poor prognostic cytogenetics with trisomy 8 as well as t, the latter not previously described in AML. Trisomy 8 in the setting of AML has been suggested to be associated with leukemia cutis, the skin infiltration of granulocytic sarcoma . Our pat


Herein we report a very rare entity of multiple hemorrhagic metastases to the brain from a primary lung choriocarcinoma in a young woman. The patient presented with recent onset of progressive headache, decreased level of consciousness and multiple episodes of vomiting. CT of the head revealed multiple hemorrhagic lesions within the brain. The patient’s serum B-human chorionic gonadotrophin was increased.  A chest X-ray revealed a right lung mass. The patient urgently underwent operative excision of the lesion in the posterior fossa, so as to prevent impending tonsillar herniation. The histology from the lesion provided the diagnosis of choriocarcinoma. After surgery, ultrasonography of the abdomen and pelvis was normal, and a chest CT revealed an enhanced and highly vascular right apical lung lesion, suggestive of lung primary choriocarcinoma, with regard to the clinical background. The patient was then started on chemotherapy, following which her serum B-HCG level decreased rapidly. This case highlights the importance of keeping this entity in the differential diagnosis of hemorrhagic lesions in any patients of a child bearing age. Early diagnosis and rapid initiation of multimodal therapy is prudent for ensuring a good outcome from an otherwise rapidly metastasizing and highly vascular lesion. Choriocarcinoma is the malignant proliferation of the syncytial cells of trophoblastic origin following gestational events, such as a term pregnancy, molar pregnancy or an abortion. We herein report one such rare case of multiple hemorrhagic metastases to the brain from primary lung choriocarcinoma in a 22 year old young woman. We also review the literature regarding primary lung choriocarcinoma and discuss recent advancements in the management of this disease.Primary lung choriocarcinoma is an extremely rare entityA 22 year old woman presented to our emergency department with a history of a recent onset progressive headache for 15 days, followed by decreased level of consciousness and multiple episodes of vomiting for the last 5 days. The patient had a history of normal vaginal delivery one month past. The patient had no history of fever, chills or any rigor associated with these symptoms, and there was no history of abnormal discharge or bleeding from the vagina. There was no other significant past medical and surgical illnesses or any relevant family history. On presentation, the patient was slightly drowsy with a Glasgow Coma Scale of E4V1M3, with bilateral pupils equal and reacting. She had bilateral sixth nerve palsies . The patient’s B-HCG decreased sharply after the first session of chemotherapy . The patient was given three cycles of chemotherapy and has been on regular follow up at the cancer centre.2. This case report discusses an even rarer phenomenon of multiple hemorrhagic metastasis in the brain from primary lung choriocarcinoma.Primary lung choriocarcinoma is a very rare entity, with < 50 cases reported currently3. This may have occurred in the present case study. Other theories discuss the probable role of primordial germ cells and the genesis of metaplasia4. Choriocarcinoma can have either a gestational or non-gestational origin6.There are various theories behind the etiology of primary lung choriocarcinoma. The foremost being embolism of trophoblastic cells during abortion, or even normal delivery, to the lung vasculature, thereby causing the cells to proliferate therein8. A high B-HCG level, as in our case, suggests a trophoblastic origin1.Sometimes large cell anaplastic carcinoma, mediastinal germ cell tumor, bronchogenic carcinoma show ectopic HCG secretion, but this elevation is mild9. Radiation has a poor response to such entity10. Therefore, the preferred therapy for gestational trophoblastic neoplasm is the EMA-CO regimen, similar to what was prescribed to our patient11.The prognosis of the condition is poor, with previous reports of a 5 year survival of <5%. However, recent advancements in chemoradiation therapy has helped to increase the overall 5 year survival rate up to 50%12. A multimodal approach is also required, constituting of neo-adjuvant chemotherapy followed by excision of the lung lesion9.The pathogenesis behind multiple hemorrhagic lesions in the brain is the tendency of such malignant trophoblastic cells to invade the vessels, and sometimes even leads to distal aneurysmsPrimary lung choriocarcinoma metastasis should be recognized as a differential diagnosis in hemorrhagic lesions of the brain, especially in patients of a child bearing age. Early diagnosis and rapid initiation of therapy is the cornerstone for a better outcome in such patients.Written informed consent for the publication of the clinical case study and accompanying images was taken from the patient. Sixth CN palsies may be better demonstrated on all directions of gaze. In the given picture, it looks like the patient also has BL ptosis!Histopathology of lung mass if included, would be better.The patient has been on follow-up at cancer center. It would be interesting to know the neurological status at follow up.1.I think the author should discuss more with data on brain metastasis of choriocarcinomaI must congratulate the author for reporting such a rare medical entity. The case has been nicely described and I am sure that this is going to be useful for other practitioners. However, I feel, few points to be considered in this case report are:I have read this submission. I believe that I have an appropriate level of expertise to confirm that it is of an acceptable scientific standard. The paper tells us “Her immune status was normal”. I think the authors should show us the date of the immune test. Since the patient was found by multiple hemorrhagic metastases to the brain, and the primary lesion from lung, how about the tumor marker from lung? Has she had a gene test of EGFR, ROS1, ALK, T790M, which could be useful if she needed target therapy? Did she need radiation for the lung tumor and when? The author need to discuss these questions.I have read this submission. I believe that I have an appropriate level of expertise to confirm that it is of an acceptable scientific standard.


Myopericytoma is a rare and usually benign tumor, which is even rarer if it occurs in the liver and stomach space. Previous reports of myopericytoma were mostly related to its pathological manifestations, while imaging reports were rare. Here, we report the computed tomography (CT), computed tomography angiography (CTA) and magnetic resonance imaging (MRI) performance for one deep myopericytoma.In this study, one deep myopericytoma in the liver and stomach space is reported. A CT that was not contrast-enhanced showed a lobulated tumor with heterogeneous density, and a contrast-enhanced CT showed that the mass had progressive enhancement. CTA showed that the blood-supply of the tumor was supplied by the anterior superior pancreaticoduodenal artery and the left gastric artery. An MRI showed the lesion had isointensity on T1-weighted imaging (T1WI) and slight hyperintensity on T2-weighted imaging (T2WI). The lesion MRI enhancement characteristics were similar to the characteristics from the contrast-enhanced CT. In this case, the enhancement pattern of the tumor was the centrifugal enhancement for both the contrast-enhanced CT and MRI. After surgical resection of the tumor, the pathological diagnosis was myopericytoma, and there was no recurrence in a short-term follow-up.The myopericytoma generally has a rich blood supply. When there is necrosis in the center lesion, the lesion has peripheral enhancement. Abdominal myopericytoma could be categorized as having centrifugal enhancement. Myopericytoma is a rare soft tissue tumor, and the concept of a myopericyte was first proposed by Dictor in 1992 . It comm2; the internal density was slightly heterogeneous, and the boundary was still clear. In CT arterial phase, the peripheral part of the mass had slight reinforcement. Gross vascular features were seen in the center of the lesion. During the portal phase and delay phase, there was progressive enhancement of lesions and reinforcement came from the center to the surrounding progress, showing a centrifugal reinforcement  desmin (+), Ki-67 , S-100 protein (−), CD34 (−), CK (−), progesterone receptor (−), estrogen receptor (−), MelanA (−), HMB45 (−), CD117 (−), and DOG-1 (−) . The routine abdominal CT and contrast-enhanced CT showed that a lobulated soft tissue mass was in the liver and stomach space with a size of approximately 7.7 × 14.2 cmAt present, the etiology of myopericytoma is unknown, and there are some cases that have reported that it may be related to trauma and virus infection , 10. Myo2 [The image findings of myopericytoma have been rarely reported, and the case studies that did report imaging findings had tumors distributed in different locations of the body , 12–16, 2 , 12–16. 2 –19 and w2 , 18. Con2 –24. In c2 , 17–20 o2 , 15, 22.2 , 16, 25.2 , 21. Whe2 . Lesions2  or lymphWT/V600E-myopericytoma should be closely followed up with because of the risk of recurrence [Surgical resection of myopericytomas has a good curative effect, and occasionally there is recurrence or rarely metastasis . In thiscurrence .The image findings for myopericytoma usually show a rich vascular lesion, and when the tumor center was necrotic, there is typically peripheral enhancement. A myopericytoma of the abdominal cavity could show centrifugal enhancement. Early resection of the tumor was necessary because it may have increased rapidly in the later stages of the disease.


Scleritis and/or uveitis sometimes accompanies patients who suffer from rheumatoid arthritis. However, few studies have reported scleritis and/or uveitis accompanying a fundus elevated lesion, such as an intraocular tumor. In this study, we report a case of rheumatoid uveitis associated with an intraocular elevated lesion.A 66-year-old female visited another eye clinic and was diagnosed as bilateral anterior uveitis, and was prescribed steroid eye drops for treatment. She had previously been diagnosed as rheumatoid arthritis at the age of 30 years. Due to vitreous opacity that appeared in her right eye, we increased the instillation of steroid eye drops and the amount of oral prednisolone. Although the inflammation had improved, anterior uveitis relapsed, and an intraocular whitish elevated lesion resembling an intraocular tumor at the superior nasal retina appeared. We speculated this lesion to be a granuloma complicated with rheumatoid arthritis. Thus, we increased the amount of prednisolone administration, and the lesion began to shrink and ultimately fully disappeared.We strongly believe that our case’s lesion was a subretinal granuloma related with rheumatoid arthritis, as it disappeared by increased corticosteroid treatment. Our findings show that we should consider rheumatoid arthritis in a differential diagnosis of such types of fundus elevated lesions. Rheumatoid arthritis is a collagen disease, and is one of the autoimmune disorders characterized by persistent synovitis, systemic inflammation, and autoantibodies . OphthalIt is thought that scleritis is caused by the immune-complex deposition, as it reportedly has been found in tissues from vasculitis in necrotizing scleritis-associated collagen disease, such as rheumatoid arthritis . Thus, iThe present case involved a 66-year-old female who became aware of decreased vision in her left eye. She had previously visited another eye clinic, and was diagnosed as bilateral anterior scleritis and prescribed steroid eye drops for treatment. Although the inflammation had subsided and the scleral redness had disappeared, vitreous opacity increased in her right eye around August 2014. Thus, she was diagnosed as uveitis and referred to the Department of Ophthalmology at Osaka Medical College in September 2014 for examination.The patient had previously been diagnosed with rheumatoid arthritis when she was 30 years of age, and she had undergone a 5 mg-per-day administration of prednisolone as a maintenance dose and long-term treatment with immunosuppressants such as tacrolimus and anti-tumor necrosis factor (anti-TNF) drugs such as etanercept by the Department of Internal Medicine at our university hospital. Her past medical history included phacoemulsification and aspiration with bilateral intraocular lens (IOL) implantation surgery in January 2014. It should be noted that there was no significant family history.Upon initial ocular examination, her visual acuity (VA) was 20/50 × S-0.25D = C-2.00D Ax90° in the right eye and 20/25 × S-0.50D = C-1.25DAx100° in the left eye, and intraocular pressure was 17 mmHg OD and 15 mmHg OS. Slit-lamp examination revealed 1+ cells in both anterior chambers, and fine keratic precipitates on both corneas. The superior sclera in both eyes showed redness and thinning of tissues Fig. . MoreoveIn regard to the follow-up treatment course, we initially increased the frequency of the instillation of steroid eye drops and added immunosuppressive eye drops. However, those drugs were ineffective, and the vitreous opacity gradually increased. Thus, we increased the amount of oral prednisolone to 20 mg per day in November 2014. Subsequently, the eye redness and vitreous opacity gradually disappeared within approximately 2 weeks, her VA slowly improved, and there was no remarkable fundus abnormality in the right eye at each examination. However, uveitis accompanied with moderate ocular pain relapsed Fig. , and funHence, we recommended to the patient that she should undergo fluorescein and indocyanine angiography examination for differential diagnosis, however, the patient wished to receive treatment without undergoing those examinations. Both the value of C-reactive protein (0.60 mg/dL) and the blood sedimentation rate (32 mm per hour) were increasing. In addition, the value of matrix metalloproteinase-3 135.0 ng/mL), an indicator of the activity of rheumatoid arthritis, was also increasing. After consultation with her rheumatologist, we increased the administration amount of prednisolone to 30 mg per day. As a result, the lesion began to shrink 1-week after, and fully disappeared 4-weeks after, initiating the increased administration  . Since iSecondly, it is vital to distinguish such an elevated lesion from an intraocular tumor, e.g., malignant melanoma, metastatic choroidal tumor, and malignant lymphoma, because those may occur with no relation to systemic disorders. However, it is sometimes difficult to distinguish between them. In our present case, we were unable to perform fluorescein and indocyanine angiography when the lesion appeared. Sin et al. reported that ocular pain is a useful symptom for differentiating nodular posterior scleritis from other forms of choroidal masses , and ourAnd thirdly, it is vital to understand that some types of uveitis may occur with a giant elevated lesion, as Sridharan et al. reported in a 53-year-old female case of posterior scleritis . In thatIn this present case, we strongly believe that the elevated lesion was granuloma caused by recurrence of uveitis. Our findings show that we should consider rheumatoid arthritis in the differential diagnosis of such types of fundus elevated lesions.


Diverticulitis is characterized by inflammation of the outpouchings of the bowel wall. Imaging findings of diverticulitis include edematous thickening of the bowel wall with inflammatory changes within the adjacent mesenteric fat. Uncomplicated diverticulitis can be treated conservatively; however, complicated diverticulitis may not be responsive to medical treatment and life-threatening conditions may occur. In this review, we aimed to illustrate the ultrasonography (US) and computed tomography (CT) features of diverticulitis and its complications including perforation, phlegmon, abscess, ascending septic thrombophlebitis (phylephlebitis), bleeding, intestinal obstruction, and fistula.Complications of diverticulitis may be highly variable.• It may be difficult to diagnose diverticulitis as underlying cause of severe complications.• MDCT is essential for the primary diagnosis of the acute diverticulitis and its complications.•  Diverticulitis is one of the most frequent bowel emergencies presenting with acute abdomen. Acute diverticulitis constitutes 3.8 % of causes of abdominal pain in patients presented to the emergency departments . ApproxiUltrasonography (US) is generally the first imaging modality used in the evaluation of acute abdomen. In diverticulitis, US demonstrates inflamed diverticulum as a noncompressible outpouching of a bowel wall with thickened and hypoechoic wall often containing an obstructive fecalith at the ostium. Adjacent bowel wall edema and thickening with edematous hyperechoic mesentery can be visualized on US . Gentle Complications of diverticulitis may be highly variable, and it may be difficult to diagnose diverticulitis as an underlying cause of severe complications. Small sized, well-contained perforations are common in the course of the disease and most cases can be managed conservatively with antibiotics and supportive medical treatment. However, unusual and more severe complications such as non-contained perforation, phlegmon and abscess, phylephlebitis, intestinal obstruction, bleeding, and fistula necessitate intensive management. These conditions should be promptly diagnosed and treated in order to prevent increased morbidity and mortality.Perforation of diverticulitis occurs secondary to severe inflammation of bowel wall layers with subsequent necrosis and loss of intestinal wall integrity. Perforation from colonic diverticulitis almost always occurs on the left side . Well-coIn the setting of perforated diverticulitis, subdiaphragmatic free air may be seen on upright abdominal X-rays. Sonographic evaluation of perforated diverticulitis performed with a high frequency linear transducer better reveals wall thickening and edema in the affected bowel segment. Diverticular perforation can be challenging to detect sonographically. Multidetector computed tomography (MDCT) is much better suited for detection of free air with a reported success rate around 85 % , 8. DireFree air resulting from perforation of diverticulitis may be detected in the retroperitoneum and mediastinum and very rarely, in the scrotal cavity . It was Diverticulitis may result in phlegmon and abscess formation. Phlegmon is detected as an inflammatory mass with heterogenous contrast enhancement adjacent to diverticulitis while abscess typically manifests as a loculated fluid collection containing air Figs.  and 4. ATreatment approach to the abscess depends on its size as well as the location and the overall medical condition of the patient. Conservative management may be preferred in abscesses less than 3 cm in size whereas percutaneous drainage or surgical intervention may be required for larger lesions . ComplicAbscess secondary to diverticulitis may also occur at distant sites such as liver, adnexa, lung and rare localizations such as brain and spine. The liver is the most common remote site of abscess formation . HematogA tuboovarian abscess may complicate acute diverticulitis of a sigmoid colon due to its close proximity to the adnexa . SonograPylephlebitis, also called ascending septic thrombophlebitis, is a rare complication of intraabdominal infections. It is typically characterized by infective suppurative thrombosis of either the portal vein or its branches, or both . DivertiThe involved mesenteric veins in pylephlebitis are closely related with the affected colonic segment. Sigmoid diverticulitis results in thrombosis of the local sigmoid vein with subsequent propagation along the IMV and the portal vein. This process is called ascending thrombophlebitis Fig.  22].Fig.Fig22].FThe clinical presentation of septic thrombophlebitis is insidious with vague symptoms, delaying early diagnosis and treatment. Therefore, a high index of clinical suspicion and appropriate use of imaging studies is of critical importance for timely diagnosis. CT is generally the first utilized modality in patients with diverticulitis with pylephlebitis. The diagnosis is generally straightforward with direct visualization of the endoluminal thrombus as a filling defect in the contrast filled mesenteric veins. In the case of portal vein thrombosis, central or peripheral hypoattenuating areas in the liver may be detected as a sign of abnormal hepatic perfusion . For theSevere intestinal obstruction in patients with diverticulitis is rare; however, partial obstruction secondary to wall edema and peripheral inflammation or abscess formation may occur. Intramuscular fibrosis seen in the chronic phase may also lead to obstruction in 10–20 % of the cases . In thesRectal diverticulitis is a very rare form of diverticulitis and may present as a rectal mass with surrounding inflammatory changes and obstructive findings. Jejunal diverticulitis, also rare, typically presents with adhesion of the epiploic band, resulting in subsequent internal hernia.Chronic diverticulitis is a variant of diverticulitis that is characterized by persisting symptoms such as abdominal pain for 6 months to 1 year and secondary obstructive symptoms. Intestinal obstruction may develop due to chronic inflammatory changes and associated dense fibrosis . SigmoidLower gastrointestinal hemorrhage can be seen in up to 5 % of patients with colonic diverticulosis . Since tOral contrast should not be used in patients with a clinical suspicion of gastrointestinal bleeding on CT as it may obscure the active contrast extravasation from the eroded vessel. Bleeding from colonic diverticulitis may be detected on unenhanced CT examinations as hyperdense endoluminal bowel content. Contrast-enhanced CT images in the arterial phase can demonstrate active extravascular contrast extravasation into the diverticulum and bowel lumen, if the flow rate of the bleeding is profuse enough. In these cases progressive contrast pooling in the bowel lumen is another confirmatory sign of active bleeding from the diverticulitis Fig. .Fig. 9BlThe rate of fistula formation is around 14 % after an episode of acute diverticulitis . They ocColovesical fistula present with free air in the bladder with thickening of the adjacent bladder wall . SeveralColouterine fistula may manifest with myometrial abscess formation . CT may Besides the usual signs and symptoms, in the course of the diverticular disease, several unusual complications may be seen. Pylephlebitis, perforation, intestinal obstruction, abscess and fistula formation may be counted among the unusual complications. MDCT is a very robust imaging tool for the primary diagnosis of the acute diverticulitis and its usual and unusual complications. Although CT is the most commonly used modality, radiologists should be familiar with US findings as sonography is generally the first preferred imaging study in the emergency department. Radiologists should be cognizant of the complex unusual complications of acute diverticulitis as they may require a multi-disciplinary treatment approach.


Appendiceal tumors are rare, late diagnosed neoplasms that may not be differentiated from adnexal masses even by advanced imaging methods and other diagnostic procedures. They may be asymptomatic and remain undiagnosed until surgery. We report a case of an 80-year-old postmenopausal woman presenting with a pelvic mass and a history of weight loss. The patient underwent laparotomy which revealed an appendiceal mucocele, for which she received a full oncological procedure. The histology report showed a low-grade appendiceal mucinous neoplasm, and the patient underwent six cycles of chemotherapy. Appendiceal tumors should be kept in mind in patients with adnexal mass. Neoplasms of the appendix are rare tumors accounting for about one percent of appendectomy specimens and 0.5 percent of intestinal neoplasms. Carcinoid tumors rank first (50%), followed by adenocarcinomas (8%) and mucinous cystadenomas of the appendix (7%) . An appeThese tumors are often discovered incidentally, either during a survey or at the time of surgery for other causes. Rupture of the mucocele, either spontaneously or accidentally during surgery, can lead to the development of pseudomyxoma peritonei, a condition in which malignant cells spread throughout the entire peritoneal cavity in the form of multiple mucinous implants on the peritoneal surfaces. The peritoneum is then seeded with mucus-producing cells, which continue to proliferate and produce mucus .It is remarkable that appendiceal mucocele is not diagnosed in almost half of cases, and it is reported to be asymptomatic in 25% of cases. The most frequent symptom that raises the suspicion is sharp or persistent pain in the right lower abdominal quadrant , 5.Herein we present a case of a postmenopausal woman with a pelvic mass and a history of weight loss. The patient underwent laparotomy which revealed an appendiceal mucocele, for which she received a full oncological procedure.An 80-year-old woman was referred to our clinic with a history of chronic abdominal pain and weight loss of 10 kilograms in the past six months. The patient was postmenopausal for 30 years and had a free medical history. Physical examination revealed no tenderness, but a hard and mobile mass palpable in the right iliac fossa. Further investigation by means of transvaginal ultrasound revealed a cystic formation, sized 83 × 65 × 64 mm in the right adnexal area. The mass was of mixed structure, comprising of solid and cystic areas. The wall was smooth and anechoic on ultrasound imaging. No papillary projections or septations were identified and there were no signs of blood flow during the color Doppler evaluation. The left ovary was illustrated measuring 25 × 13 × 11 mm, but the right ovary could not be visualized Figures . A polypThe patient underwent laparotomy with the diagnosis of a pelvic mass, in which a cystic tumor originating from the appendix, sized 80 × 90 mm, was identified, whereas the uterus and ovaries were atrophic. The abdominal viscera were covered with surgical pad gauzes to protect from spillage of the cyst contents. However, several intestinal loops adhered to the cyst, and despite meticulous dissection, the cystic mass ruptured intraoperatively. Frozen section was performed, which revealed malignant mucinous neoplasm. Appendectomy, omentectomy, total abdominal hysterectomy, and bilateral salpingooophorectomy were performed, in collaboration with the General Surgery Team of the hospital. The histopathologic examination of the surgical specimen revealed a low-grade appendiceal mucinous neoplasm with focal invasion of the muscle layer , without desmoplastic reaction. The patient had an uneventful recovery and was discharged from the hospital one week later. Soon afterwards she underwent six cycles of chemotherapy and up to present, 12 months after surgery, she remains in good health.Appendiceal mucocele is considered very rare and usually presents as a distended, mucus-filled appendix. The course and prognosis of appendiceal mucocele is related to the histologic subtype. This tumor appears to have a slight female predominance and is usually diagnosed in patients in their fifth and sixth decades of life; however it may occur at any age , 6. NeopPreoperative diagnosis of an appendiceal mucocele is difficult to accomplice because of the lack of specific symptoms. An appendiceal mucocele should be suspected in elderly women with the atypical finding of a mass originating from the adnexa in ultrasonography. This tumor may also represent an incidental finding during radiologic or endoscopic evaluation of nonspecific complaints. Elevated levels of tumor markers  have been reported .An abdominal CT scan can provide useful findings, such as a round or tubular cystic mass with calcification at the expected site of the appendix. We can also presume the existence of the tumor based on colonoscopic findings  , 12. HowAppendicitis, a mesenteric or duplication cyst, and an adnexal mass are to be considered in the differential diagnosis. In our case a diagnosis of a cyst originating from the right adnexa was initially established; however, an appendiceal mucocele was evident only at laparotomy. Perforation of the appendix or mucous extravasation at the time of surgery may happen in one-third of patients and this may lead to pseudomyxoma peritonei . The proOpen surgical resection constitutes the treatment of choice, even for a benign-appearing appendiceal mucocele, since lesions that appear to be benign on imaging studies may harbor a cystadenocarcinoma. Laparoscopic treatment should be avoided, as there is increased risk of rupture. In case of malignancy, routine oophorectomy should be performed at the time of surgery, since the ovaries represent a common organ for metastases , 17. If In conclusion, appendiceal mucocele is a rare finding in abdominal surgery. Ultrasonography and computerized tomography represent useful tools for diagnosis. However, diagnosis is often intraoperative and is based on histopathological examination. The course and prognosis of this rare tumor is related to the histologic subtype. Retention cysts, mucosal hyperplasia, or cystadenomas present with excellent survival rate (91 to 100%) after standard appendectomy. In patients with appendiceal cystadenocarcinomas, five-year survival has a wide range (6 to 100%), based on stage .