Laparoscopic cholecystectomy (LC) carries a significant risk of gallbladder perforation and resulting scattering of bile stones into the abdominal cavity. The retrieval of the spilled stones is not always possible by laparoscopic technique. Most cases do not create long-term problems, although some cases may be complicated in future and create uncertainties regarding the correct diagnosis. Diagnosis can be difficult, and in most cases the patient may require open surgery for management of these complications. Herein, we report a case of acute abdomen due to spilled stones occurring 10 years after LC. In the first stage, definitive diagnosis could not be made with computed tomography examination. Finally, the patient was diagnosed with explorative laparotomy. Laparoscopic cholecystectomy (LC) has become the standard treatment for symptomatic cholelithiasis and acute cholecystitis. However, LC is associated with some complications that are rare in open cholecystectomy . Gallbla3, hemoglobin: 11.9 g/dl, C-reactive protein: 24 mg/dl, and other biochemical parameters were also normal. On oral contrast-enhanced abdominal computed tomography (CT) performed in the emergency room (A 42-year-old woman presented to our emergency department with a painful swelling in the suprapubic region persisted for 3 days. She had a history of LC 10 years back at our center for symptomatic gallstones and had no history of comorbidities. The patient had a history of cesarean section twice. She had a pulse rate of 92/min, blood pressure of 110/60 and a temperature of 37.4°C. Physical examination revealed abdominal tenderness in the suprapubic region, right and left lower quadrant, and exhibited signs of peritoneal irritation, muscle guarding and rebound tenderness. Laboratory tests resulted with white blood cell: 15 200/mmncy room , the mesncy room . These sDespite the rare nature of gallbladder perforation and resulting scattering of bile stones into the abdominal cavity, they may still cause diagnostic challenges and substantial morbidity in both early and late postoperative periods. Complications due to peritoneal gallstones after LC are infrequent with a rate of 1.7 complications per 1000 cases . The repStone spillage from gallbladder occurs during gall bladder extraction, by tearing with grasping forceps or during dissection of the gallbladder from the liver bed. Predisposing factors for gallbladder perforation and stone spillage include acute cholecystitis, pericholecystic adhesions, obesity and male sex. The problem of stone spillage is aggravated by spread of calculi because of peritoneal irrigation and pneumoperitoneum .The reported complications due to gallstone spillage include peritoneal–cutaneous sinus tracts, intra-abdominal abscess, liver abscess, subhepatic inflammatory mass, persistent discharging trocar sites, micro abscesses, retroperitoneal abscess, granuloma, cholelithoptysis, small bowel obstruction, small bowel fistula, colonic fistula, bowel perforation and ileus as a result of sticking of small intestine inserts into the abscess wall. Bile stones may remain silent in the abdominal cavity for a long time until a septicemic focus causes calculus to become infected and produce abscesses .Although early complications often occur in the form of peritonitis or abscesses, late complications usually occur as a result of migration of the stones. The organism tries to throw the stone out of it by accepting it as a foreign body. Generally, complications require surgical intervention. Such patients should be well informed after surgery, and stone spillage should be well documented in order to avoid medico-legal issues and diagnostic dilemmas in the future. In our case, no documentation or surgical notes about previous surgeries were found. Complications due to gallstone spillage may take months or years to present. Ultrasound, CT or magnetic resonance imaging can be used to detect a spilled stone in suspected cases presenting late after cholecystectomy .Radiological interventions, laparoscopic methods and open surgery are available to remove such stones from the abdominal cavity and drain any abscess if present. Abscess puncture and aspiration without stone removal should be avoided because these often result in recurrence. An observational study involving 82 surgeons revealed that conversion to an open procedure was performed only in 3% of patients in whom gall stones were spilled during LC. Although removal of spilled gallstones is not recommended for all patients, an attempt at removal should be performed whenever possible . Our caset al. [Brockmann et al.  reportedDuring LC, it is common to encounter gallbladder perforation ending up with spillage and loss of some gallstones. It is important to attempt to retrieve spilled gallstones. Though uncommon, these stones may lead to early or late complications, which can be a diagnostic challenge and cause significant morbidity to the patient.None declared.Concept: M.T.K., İ.Ç.; Design: M.T.K., U.A., Ö.B.; Supervision: M.T.K., Ö.B., H.B.; Fundings: M.T.K., İ.Ç.; Materials: M.T.K.; Data collection and/or processing: İ.Ç.; Analysis and/or interpretation: M.T.K., Ö.B., H.B.; Literature review: M.T.K., U.A., H.B.; Writing: M.T.K.; Critical review: M.T.K., U.A.Written informed consent was obtained from the patient for the anonymized information to be published in this article.


Supratentorial hemangioblastomas have rarely been described in the literature. Pituitary stalk hemangioblastomas are extremely rare and almost always are associated with von Hippel Lindau disease. Herein, we report a sporadic case of pituitary stalk hemangioblastoma in a 36-year-old male and review the current literature regarding this pathology. In our case, complete resection of the lesion was achieved using the transglabellar approach. Hemangioblastomas are histologically benign tumors of vascular origin that are usually cystic, occur in the cerebellum, and represent approximately 2% of all intracranial tumors -3. SupraWe present a 36-year-old male who was admitted to our department due to the gradual worsening of the left eye vision. Ophthalmologic evaluation of the patient’s retina showed no remarkable ﬁndings while ocular examination showed visual acuity 8/10 on the right eye and 1/10 on the left eye. The formal visual field testing, by using the standard Goldman perimetry study, revealed a left upper temporal quadrant deficit. Further neurological examination was normal. No endocrine dysfunction was identified preoperatively. Brain computed tomography (CT) scan revealed a sellar mass with sellar enlargement and dorsum sella erosion Figure -4.The patient underwent a surgical operation via a transglabellar approach Figures -6. A higHistopathological examination of the tumor showed a cellular proliferation of stromal cells, interlaced by numerous thin-walled vessels, foam, and vacuolated histiocytes. No mitoses were identified. In histochemical and immunohistochemical stains, stromal cells were positive to vimentin and epidermal growth factor receptor (EGFR) and in limited locations, also positive in EMA and neuron-specific enolase (NSE) while negative in CK7, CK20, CK 5/6, glial fibrillary acidic protein (GFAP), pancytokeratin, CK34βΕ12, S-100, and α-inhibin. The cellular proliferation index ki67 was less than 1%. Differential diagnosis included angiomatous meningioma, angioblastic meningioma, transitional angioblastic, and angioblastomatous meningioma. The overall histological diagnosis was capillary hemangioblastoma  Grade I).Postoperatively, the patient had considerable improvement in his visual acuity and temporal quadrant deficit. Except for transient diabetes insipidus, the rest of the postoperative course was uneventful, including any endocrine dysfunction.After the histological diagnosis, initially, VHL syndrome was excluded via further imaging studies, including ophthalmic ultrasonography, abdominal, cervical, thoracic, and lumbar MRI. Although imaging studies were negative for other tumors and there was no known family history, genetic screening was suggested to the patient for von Hippel-Lindau gene mutations. No residual tumor was recognized in the brain MRI six months after surgery and the genetic test was negative for VHL mutations Figure .Hemangioblastomas are benign lesions that originate from the vascular system and have been reported in a variety of locations in the central nervous system. These tumors present as either isolated or multiple lesions. Isolated lesions occur in 80% of patients with VHL syndrome, and in 95% of patients without this disease. They are usually found in the cerebellum, but they have also been reported in the spinal cord, the brain stem, and, in rare cases, in the cerebrum. Typically, patients in the fourth and fifth decades are affected, but there are also reports of congenital lesions. There are only 132 previous reports in the literature of supratentorial hemangioblastomas . ReportsVon Hippel-Lindau disease is positively correlated with supratentorial hemangioblastomas when compared with non-supratentorial central nervous system hemangioblastomas, particularly when present in the sellar/suprasellar region . PituitaSurgical resection (open or endoscopic) of these lesions is necessary when they become symptomatic, usually presenting with visual defects and endocrine dysfunction. Due to their high vascularity, open craniotomy is generally preferred in order to prevent uncontrolled bleeding and partial excision, but this approach results more often in pituitary stalk excision as compared to the transsphenoidal approach ,9,16. ToOur study reports the ninth case of sporadic pituitary stalk hemangioblastoma, in which total resection was achieved by the transglabellar approach that is not so commonly used. Preoperative imaging should be thorough regarding highly vascularized lesions and angiography should be generally included in the preoperative setting, as it can be helpful not only as a diagnostic tool but also as a treatment modality with the embolization of large feeding vessels in cases of hemangioblastomas. Large multicenter studies are necessary to evaluate the role of radiotherapy as a primary or adjuvant treatment.


A thyroglossal duct cyst (TGDC) is a congenital malformation in the neck. Surgical management is often recommended due to risk of recurrent infections and rare possibility of malignancy.Herein, we describe the case of an adult presenting with tender neck mass and fever. She had a history of previous surgical excision of her TGDC as a child. On evaluation she was found to have a recurrent TGDC complicated by acute infection via computed tomography imaging.In patients who have had previous surgical intervention to remove a TGDC, recurrence with infection should remain a diagnostic consideration. Thyroglossal duct cysts (TGDC) are a common congenital malformation, typically presenting in the pediatric population. Predominant occurrence is in the first decade of life with physical exam findings of a midline mobile mass at the level of the hyoid.A 24-year-old previously healthy female presented to the ED with four days of fever, sore throat, neck swelling, and voice change. She had been previously evaluated at an urgent care facility where she had a negative rapid antigen detection test for group A streptococcus, but was referred to the ED for suspected peritonsillar abscess. Her past medical history was significant for tonsillectomy, adenoidectomy, and TGDC excision. On physical exam, she was appropriately managing her secretions, protecting her airway, and was able to lay supine without experiencing any respiratory distress. Her neck was diffusely swollen, with tender submandibular and anterior cervical lymphadenopathy. Her voice was notably muffled. No discrete fluctuant mass could be palpated or visibly appreciated. The remainder of her physical exam was unremarkable and her vital signs were within normal limits.Computed tomography (CT) of the neck with contrast revealed a 1.0 × 1.3 × 2 centimeter midline lobulated fluid collection with mild rim enhancement immediately anterior to the lingual tonsils . The otoThe differential diagnosis for neck swelling, and voice change includes infectious etiologies, oncologic processes, lymphadenopathy, and various cysts. As previously mentioned, the majority of TGDCs will present in childhood, with 60% before age 20. While infrequent, primary occurrence can also present in adulthood, with an even distribution between males and females.3Recurrence rate following excision remains uncommon. The earlier in childhood the excision is performed, the higher the rate of recurrence.1Although infection of a recurrent TGDC is rare, early recognition and diagnosis in the ED is key to appropriate dispositioning of patients, and avoidance of potential airway compromise.What do we already know about this clinical entity?Thyroglossal duct cysts (TGDC) commonly present in childhood as benign midline neck masses, usually treated with surgical resection.What makes this presentation of disease reportable?This is the first case report detailing a recurrent TGDC complicated by acute infection.What is the major learning point?Acute infected TGDC should remain on the differential diagnosis for neck swelling regardless of history of previous excision.How might this improve emergency medicine practice?As emergency clinicians it is important to keep common childhood pathologies and their complications on the differential even into adulthood.


Idiopathic deep venous thrombosis is a relatively common clinical finding. The aim of this article is to report and discuss a case of lymphoma presented as DVT in line with SCARE guidelines with brief literature review.A 19-year-old male presented with right lower limb swelling for two week duration. Examination showed pitting leg edema with warm tense calf. Hematological investigations showed lymphopaenia. Duplex ultrasound showed DVT of right common femoral vein with inguinal lymphadenopathy. Computed tomography showed inguinal lymphadenopahthy. Histopathological examination of lymph node specimen confirmed the diagnosis of Non-Hodgkin’s Lymphoma (Burkett's type).Several series have documented a significantly higher risk of malignancy in patients with presumed idiopathic DVT. Among such patients, 7.6% have been noted to have a malignancy during follow-up; the incidence of occult malignancy diagnosed within 6–12 months of an idiopathic DVT is 2.2–5.3 times higher than that expected from general population estimates.Idiopathic DVT regardless of age should be approached seriously with a special consideration to malignancy, lymphoma could present only with DVT. The acute onset of unilateral leg edema points mostly to a diagnosis of thrombophlebitis, especially with the presence of inﬂammation such as hotness, tenderness and erythema . A more The aim of this article is to report and discuss a case of lymphoma presented as DVT in line with SCARE guidelines with brief literature review .1.1A 19-year-old male presented to the outpatient clinic of vascular surgery complaining of gradually increased swelling involving his right lower limb (RLL) for two week duration. He was an accountant at a grocery shop with clear past medical and past surgical history.1.22 with normal vital signs and not feverish. Examination of RLL showed pitting leg edema grade 2/4 with warm tense calf with 3.5 cm discrepancy with the other leg. Distal pulses were positive. Neurological examination was normal. Also there was discrete rubbery non-tender right inguinal lymphadenopathy of variable size of 1–4 cm.Clinical examination revealed a well-built man with body mass index of 25.8 kg/m1.3Hematological investigations showed lymphopaenia. Duplex ultrasound of RLL showed DVT of right common femoral vein with inguinal lymphadenopathy; largest one was 4 cm in size with an ill-defined mass 6 cm in size above the inguinal ligament encasing both external iliac artery & vein.IV contrast enhanced CT-scan of abdomen & RLL showed enlarged lymph nodes encasing external iliac artery & common femoral artery. Excisional biopsy was planned & performed successfully under general anesthesia. Histopathological examination of the specimen confirmed the diagnosis of Non-Hodgkin’s Lymphoma (NHL) (Burkett’s type).1.4The patient was advised to have RLL bandaging, I.V anticoagulant and referred to hematological department for further management.2Malignancy, surgery and trauma within the preceding three months remain the most significant risk factors for outpatient DVT, approximately 47% of outpatients with a documented DVT have one or more of the recognized risk factors, approximately 20% of all first-time acute venous thromboembolism i.e. DVT or pulmonary embolism (PE) events are associated with malignancy, an estimated 1 in 200 individuals with malignancy will develop either DVT or PE, a fourfold higher risk than those without malignancy [Several series have documented a significantly higher risk of malignancy in patients with presumed idiopathic DVT. Among such patients, 7.6% have been noted to have a malignancy during follow-up; the incidence of occult malignancy diagnosed within 6–12 months of an idiopathic DVT is 2.2–5.3 times higher than that expected from general population estimates .The most prevalent clinical presentation of NHL is inguinal or cervical lymphadenopathy. In many cases, patients notice masses in various part of the body and look for a physician’s advice. Generally, lymph nodes in case of lymphoma are non-tender, firm, and does not associated with a locao-regional infection. In other cases, lymphnode enlargement are found in more critical areas like the retroperitoneum or mediastinum leading to compression symtoms. Cough, chest pain, superior vena cava syndrome, abdominal pain, spinal cord compression, back pain and ureteral compression leading to symptoms of renal insufficiency are characteristics . ElgendyRonsdorf et al. searched 236 patients with proven DVT for occult cancer and found eleven cases. They concluded that the prevalence of occult malignancy in idiopathic DVT was higher than previously thought. They recommended that taking history, proper clinical examination, requesting simple laboratory tests with chest x.ray are the mandatory strategies that should never be forgotten when one deals with DVT. In their opinion, abdominal ultrasound adds nothing to the work up and can be omitted .The investigators confirmed that vascular compression by the surrounding enlarged lymph nodes is the most significant risk factor for development of DVT in case of lymphoma . The curIn addition, concerning the concept of Virchow’s triad which was named after the German pathologist, Rudolf Virchow, where he observed that thrombosis occurred when the following three situations were met; vascular wall  injury, stasis of blood, and changes in the consistency of blood that enhances coagulation (hypercoagulability) with concerning stasis which occurs as a result of external compression of the vessel wall which eventually leads to thrombos formation as in the present case, other studies have supported this concept; Hisatomi et al. reported a case of DVT associated with iliac lymph node metastasis as a result of a primary unknown tumor ,10. ChiaIn this report, it will be clear that extensive search for underlying causes in case of DVT is of paramount to exclude a serious disease.In conclusion, idiopathic DVT regardless of age should be approached seriously with a special consideration to malignancy, lymphoma could present only with DVT.There is no conflict to be declared.No source to be stated.Approval is not necessary for case report in our locality.Consent has been taken from the patient and the family of the patient.Okba F. Ahmad: Surgeon managing the case and follow up, writing the manuscript.Shvan H. Mohammed, Fahmi H. Kakamad, Berwn A.Abdulla and Diyar A. Omar: Writing the manuscript and follow up.Abdulwahid M. Salih, Mohammed Q. Mustafa, Rawezh Q. Salih, Hunar Ali Hassan, Suhaib H.Kakamad, Hawbash M. Rahim, Jaafer O. Ahmed: literature review, final approval of the manuscript.Not applicable.Fahmi Hussein kakamad.Not commissioned, externally peer-reviewed.


Squamous cell carcinoma (SCC) of the colon is an extremely rare condition, and its pathogenesis is not fully understood. Bowel perforation is a very infrequent manifestation of colonic SCC, and only a few cases have been reported in the literature involving sigmoid and splenic flexure perforation. To the best of our knowledge, no cases of ileocecal perforation have been documented in the literature. Here we present a case of cecal SCC that presented with bowel perforation, necessitating emergent surgical intervention. Histopathological examination showed SCC with lymph node metastasis. This case reviews current knowledge and highlights the rare manifestation that these rare tumors can present. Primary squamous cell carcinoma (SCC) of the colon is an extremely rare entity accounting for only 0.1-0.25/1000 of all colorectal malignancies [The clinical presentation of colonic SCC is described to be similar to that of colorectal adenocarcinoma; however, bowel perforation is an unusual presenting manifestation. To establish the diagnosis of primary colonic SCC, other primary tumors must be excluded as part of the diagnostic criteria. Diagnostic criteria also include histological confirmation and lack of continuity of the tumor with anal epithelium or squamous lined fistula .Currently, there are no treatment guidelines for this condition. Surgical resection has been commonly used with the aim of complete excision with negative margins. More recently, in addition to surgery, chemotherapy has been used for the treatment of this condition. SCC of the colon has been reported to have higher mortality rates than colonic adenocarcinoma . Here weA 61-year-old African American female presented to the emergency department with a two-day history of worsening right lower quadrant pain and melena. She also reported 20 lbs of unintentional weight loss and intermittent fever. She had no personal or family history of malignancy. On physical examination, her abdomen was distended and diffusely tender to palpation. A CT scan of the abdomen and pelvis showed intraperitoneal free air Figure  and asciThe pathological gross description showed two segments of intestine adherent to one another by a perforated mass. The mass was white-gray, necrotic, firm, and measured 8 cm x 6 cm x 4 cm Figure . MicroscEntamoeba histolytica. A history of previous surgical procedures or radiation therapy has been found in patients with colonic SCC [SCC of the gastrointestinal tract is common in the esophagus and anus; however, it rarely affects the colon and rectum. The first case of SCC of the colon was described in 1919 . Since tonic SCC . Hicks aonic SCC . Our patThe clinical manifestation of primary SCC of the colon has been described to be comparable to colorectal adenocarcinoma. Frequently reported symptoms include rectal bleeding, abdominal pain, change in bowel habits, weight loss, and bowel obstruction . In the The rarity of these tumors and the lack of randomized controlled trials impede the creation of treatment guidelines. Generally, surgical intervention has been routinely performed with the more recent use of neoadjuvant chemotherapy (5-fluorouracil and mitomycin C) and radiation therapy. The overall five-year survival rate for these patients is 30%-35%, which is less than those with adenocarcinoma of the colon , 10. If Primary SCC of the colon is a rare entity, and its pathogenesis is not fully understood. Given the small number of cases reported, there are no established treatment protocols for this condition. Surgical intervention has been routinely employed in addition to adjuvant chemoradiation and followed by close endoscopic surveillance. Documentation of this case is crucial as it presented in an unusual manner with bowel perforation. We hope that our experience can help further understand this condition and help in the creation of clinical guidelines in the future.


Introduction. Adrenocortical carcinomas (ACCs) are infrequently reported to present with severe hypoglycemia syndrome resulting from the secretion of insulin-like growth factor II (IGF-II) by tumor cells. Adrenocorticotropic hormone- (ACTH) independent hypercortisolism is the norm of hormonally active ACCs, but aberrant ACTH production by tumor cells can theoretically cause ACTH-dependent hypercortisolism. The purpose of this report was to present a case of an ACC manifested with the co-occurrence of two extremely rare presentations. Case Description. We present a rare case of a 43-year-old male patient admitted with recurrent episodes of severe non-ketotic and non-insulin-mediated hypoglycemia due to IGF-II mediated disease and ACTH-dependent Cushing's syndrome. He was diagnosed with a diffusely disseminated adrenocortical carcinoma with immunohistochemistry of tumor cells showing focal ACTH immunostain positivity. Conclusion. Non-islet cell tumor hypoglycemia and ACTH-dependent Cushing's syndrome are extremely rare presentations of an ACC, and co-occurrence of these entities in a single patient is never reported in the literature. Adrenocortical carcinomas (ACCs) are rare but aggressive tumors which show an incidence of 1-2 cases per million populations per year  in assocA 43-year-old Sri Lankan male presented to a tertiary care hospital of Sri Lanka (SL) in January 2019 with recurrent episodes of early morning dizzy spells of six-month duration and a history of resistant hypertension and intra-abdominal malignancy for which he received several types of treatment abroad and locally until 3 months before.He started to experience recurrent episodes of early morning dizzy spells which were associated with excessive hunger, sweating, and palpitations that were resolving with a sugary drink or food in July 2018. These episodes were confirmed to be hypoglycemia at a local hospital with positive Whipple's triad while awaiting surgery for the aforementioned malignancy. There was some improvement of these symptoms after resection of the tumor; hence, no further evaluation was performed. However, hypoglycemia symptoms recurred in September 2019 and gradually progressed thereafter with increasing frequency and severity, up until 2 weeks, when he started to develop episodes of disorientation and confusion, which prompted him to seek medical attention. There were no episodes of coma or seizures to report, and he denied symptoms of overt liver or renal failure apart from the loss of appetite. He neither had features of clinical depression nor history to suggest abuse of oral hypoglycemic agents, insulin, recreational drugs, or alcohol. He was off from any medication over 3 months before the admission.+ 2.8 mmol/l), normal blood counts, renal, and liver functions. He was noted to have heart failure with an ejection fraction of 38% with a concentric left ventricular hypertrophy. His abdominal ultrasonography and subsequent whole-body PET/CT had shown a left suprarenal mass of 22 cm × 13 cm × 23 cm in size with coeliac and para-aortic lymphadenopathy (maximum diameter of 3 cm), and hepatic, lung parenchymal, and subpleural metastasis. Biopsy of abdominal mass had shown tumor cells with a high degree of anaplasia, and immunohistochemistry was compatible with probable adrenocortical malignancy. He had received 3 cycles of chemotherapy according to the records, but no details were mentioned regarding the types or names of medications.His past medical history revealed poorly controlled hypertension since mid-2017 which was not properly controlled as he went for employment abroad. According to his medical records, he admitted to a hospital abroad in January 2018, with subacute onset moderate to severe retrosternal chest pain with difficulty in breathing. Additionally, he complained of progressive abdominal distension, nonspecific abdominal pain, significant loss of appetite, loss of weight of 2 months, and chronic watery stools without blood or mucus discharge of similar duration. He also had recurrent nonspecific headaches and palpitations but no episodes of flushing. Examination findings on admission recorded pulse rate (PR) of 96 beats per min with a blood pressure (BP) of 170/100 mmHg. Laboratory workup showed hypokalemia  scan of the chest, abdomen, and pelvis showed previously detected heterogeneously enhancing left suprarenal mass with central necrosis and foci of calcifications without significant interval change in size. There were multiple liver, lymph node, and pulmonary metastasis to note .He underwent resection of abdominal mass in July 2018 and made an uneventful and complete recovery after the surgery with improvement in symptoms of hypoglycemia. However, histology of the tumor cells showed a high degree of anaplasia and basic immunohistochemistry pattern was more in favor of a malignant pheochromocytoma. Nevertheless, the patient defaulted from follow-up diverting back to Ayurveda treatment without waiting for further evaluation and management.He had no significant family history of malignancy or chronic diseases to note. He was a social drinker and a non-smoker.2. His PR was 96 beats per minute, and BP was 180/90 mmHg. He had large tender hepatomegaly and a well-healed midline abdominal scar. The rest of the clinical examination was unremarkable with no edema, generalized pigmentation, or other discriminatory features of Cushing's syndrome except for significant proximal myopathy.On examination at our center, he was seemingly well with a BMI of 25.2 kg/mDuring the hospital stay, his vital parameters were stable, and the lowest recorded blood sugar level was 15 mg/dL with symptoms. His laboratory workup revealed neutrophil leukocytosis, anemia, mildly elevated transaminases, and normal renal functions .Further biochemical evaluation of persistently low blood sugar was favoring non-ketotic and non-insulin-dependent hypoglycemia . ConsideMoreover, CECT chest, abdomen, and pelvis showed recurrence of the previously detected tumor  with multiple liver (largest of the metastases being 12 × 11 cm in size in segment VII and VIII), renal (40 × 62 mm), and lung metastases . But theAdrenal hormone analysis was performed as the clinical picture was suggestive of a biologically active tumor, in addition to the dilemma created by the initial histology report . The horWe reassessed tumor tissues in parallel to the above evaluation with extended immunohistochemistry (IHC) profiles. Histology  showed aThe IHC staining profile  showed pAs the patient had ACTH-dependent Cushing's syndrome with a theoretical possibility of tumor cells secreting ACTH, we included ACTH immunostain to the IHC profile. Tumor cells showed granular cytoplasmic positivity for ACTH favoring aberrant ACTH production by ACC cells .Alleviation of the troublesome hypoglycemia to improve quality of life was a priority of this patient's management. He was offered frequent meals and intermittent oral dextrose but failing that commenced on continuous intravenous dextrose infusions with frequent monitoring of blood sugar. Oral diazoxide seemed not effective. Subsequently, the patient was commenced on subcutaneous recombinant growth hormone (rhGH) 3 mg/daily and rapidly increased up to 4 mg/daily for which he showed a significant reduction of severity and frequency of hypoglycemia, hence could limit dextrose infusions. After extensive discussions with the oncologist, patient, and the family, he was opted for palliative chemotherapy believing that reduction of tumor burden to some degree would further help to alleviate symptoms of hypoglycemia. Unfortunately, the patient succumbed to the illness while receiving palliative chemotherapy.This patient was diagnosed to have an aggressive, functional adrenocortical carcinoma with diffuse metastasis, which was complicated with paraneoplastic severe NICTH and ACTH-dependent Cushing's syndrome resulting from aberrant ACTH secretion.This patient had hypercortisolism, hyperandrogenemia and negative urine metanephrines in the presence of disseminated adrenal malignancy favoring a tumor of adrenocortical origin. But there was a misclassification of the tumor in the initial histology report. The considerable difficulty associated with differentiation between adrenocortical (ACC) and medullary (pheochromocytoma) tumor with a high degree of anaplasia was previously noted in the medical literature . Even thNICTH is a rare paraneoplastic syndrome that is only second to insulinoma as a cause for tumor-induced hypoglycemia . This isDefinitive management of ACC is the mainstay of therapy for NICTH after the acute management of hypoglycemia, which seemed impossible to achieve with the extensive tumor burden of this patient. Glucocorticoids , 11, gluFurthermore, non-ACTH-dependent Cushing's syndrome is the norm of adrenal-mediated Cushing syndrome commonly seen in association with hormonally active ACC. However, our patient had biochemically confirmed Cushing's syndrome with non-suppressed serum ACTH levels. ACCs were never reported to be associated with ectopic ACTH production in the medical literature thus far. But, we hypothesized that this could be a possibility since there was no obvious clinical or imaging evidence to suggest pituitary disease or other ectopic focus. This hypothesis was proved by the immunopositivity of tumor cells for ACTH, indicating aberrant ACTH production.Non-islet cell tumor hypoglycemia is an extremely rare presentation of ACCs. Though non-ACTH-dependent hypercortisolism is the norm of ACC, it can be associated with ACTH-dependent Cushing's syndrome with aberrant ACTH production by tumor cells. To the best of our knowledge, the co-occurrence of these presentations associated with ACC is never reported in the literature. Increased level of suspicion and broad-minded approach in the evaluation of this type of rare endocrine malignancies would help in diagnosing its rare presentations.


Ventral incisional hernia is a common problem after abdominal surgery. Most patients with these hernias present with greater omentum and gastrointestinal prolapse. However, hepatic herniation through a ventral incisional hernia is a rare phenomenon that has been seldom reported in the literature. We report the case of a ventral incisional hernia with hepatic herniation treated with laparoscopic repair.A 68-year-old Japanese women with a history of myocardial resection for hypertrophic cardiomyopathy 1 year earlier was admitted to our hospital with symptoms of vomiting and epigastric pain. Physical examination showed a 4-cm epigastric mass. Abdominal computed tomography revealed left hepatic lobe herniation through the lower edge of a mid-sternal incision. We diagnosed the patient with a ventral incisional hernia with hepatic herniation. The patient underwent laparoscopic hernia repair. During an 18-month follow-up, no recurrence or symptoms have been observed.To the best of our knowledge, this is the first case report of laparoscopic repair of ventral incisional hernias with hepatic herniation. Laparoscopic repair was useful and suitable for this rare herniation due to its minimally invasive nature and ability to achieve sufficient visibility of the surgical field. Laparoscopic repair could be a potential treatment option for elective surgery for this disease, which is often treated conservatively. Ventral incisional hernias are a common problem after abdominal surgery . Most pa2) and fatty liver, laboratory test results demonstrated normal hemoglobin and hematocrit levels, normal liver enzymes, and normal renal function. Physical examination revealed a 4-cm painless mass protruding through the lower edge of a mid-sternal incision, which gradually expanded 3 months after the cardiac surgery. Abdominal computed tomography revealed left hepatic lobe herniation through a subxiphoid incision (Fig. A 68-year-old Japanese women presented to our hospital with vomiting and epigastric pain. Her medical history included left ventricular myectomy with median sternotomy for hypertrophic cardiomyopathy 1 year earlier. No abnormal vital signs or cardiovascular function were found. Although she suffered from obesity (body mass index, 37.3 kg/mThe case reported herein has two important clinical implications: hepatic herniation can occur within a ventral incisional hernia, and laparoscopic repair was useful in the treatment of this type of hernia. Our case is the first report of laparoscopic repair of a ventral incisional hernia with hepatic herniation.First, prolapse of parenchymal organs such as the liver is rarely found in a ventral incisional hernia. In most cases, the contents of a ventral hernia include the small intestine, colon, and fat or fibrous tissue . AccordiSecond, this case demonstrated that laparoscopic repair was useful for a ventral incisional hernia with hepatic herniation. A review of the literature revealed no standard treatment strategy for hepatic hernia. Generally speaking, surgical treatment is selected when conservative treatment becomes ineffective. Out of the ten cases of hepatic herniation mentioned previously, four were treated with non-laparoscopic surgery. In open surgery, positioning of the mesh may be carried out through a variety of methods such as onlay, sublay, or underlay. However, laparoscopic repair is based on an underlay approach, which serves to visually reinforce a substantial portion of the abdominal wall compared with open procedures . TherefoOut of the five cases of subxiphoid hernia with hepatic herniation mentioned previously, four were obese women with fatty liver , 6, 7. LIn conclusion, hepatic herniation can occur within a ventral incisional hernia, and be repaired laparoscopically by suturing the mesh to the falciform ligament and diaphragm. Although special consideration must be taken into account in patients with previous heart disease and/or hernias following sternotomy, this surgical method is considered suitable due to its minimally invasive nature and increased visibility of the surgical field.


Herein, we report a rare case of a stress fracture through a pre-existing non-ossifying fibroma (NOF) of the femur of a 12-year-old child that raised suspicion of a malignancy. Although NOFs are very frequent, in the vast majority of cases, they are completely asymptomatic. When encountered in a painful area, especially if combined with atypical radiographic features, they may mimic a malignancy. We discuss the radiographic findings, the differential diagnosis, and the relevant points of the patient's history that helped to establish the diagnosis. Benign and non-neoplastic conditions can mimic malignant bone tumors in imaging studies. Usually, each of them separately does not pose a significant diagnostic problem, like a non-ossifying fibroma (NOF) and a stress fracture. When they exist simultaneously, however, potential diagnostic difficulties can increase, especially when faced by a physician who is less experienced in musculoskeletal pathology.In those cases, the correct diagnosis could be established after a careful study of the patients’ history and clinical status, together with different imaging modalities. After precise evaluation, a correct diagnosis and plan for treatment could be made ,2. StresA 12-year-old boy was referred to the orthopedic department with a suspected malignancy in the left femur. He had had exertional pain in the left thigh for several months. After a fall four days before the visit, pain became constant and sharper and especially severe with weight-bearing. On clinical examination, no palpable mass was detected but only a tender point over the distal thigh. In the distal femoral diaphysis, radiography revealed a large eccentric heterogeneous area with somewhat dulled osteosclerotic margins oriented along the axis of the femur consistent with the diagnosis of an NOF. Surprisingly, the overlying periosteum showed atypical interrupted lamellar reaction over a short distance of the near cortex and longer non-interrupted periosteal reaction over the far cortex that gave the lesion a somewhat sinister appearance Figure . CarefulIn the current literature, NOF has been reported as the most common metaphyseal benign fibrous lesion of the long bones. Usually, it is asymptomatic and incidentally found on radiography ,6. It tyA stress fracture is usually transverse and could be detected on plain radiographs; however, CT reveals it better, especially in cases of a longitudinal fracture line that could be difficult to establish on a radiograph . CT alsoAccording to Easley and Kneisl 1997), lesions affecting more than 50% of the bone diameter could predispose to fracture . It shou97, lesioIn cases of an interrupted lamellar periosteal reaction that is generally viewed as a worrisome radiographic feature, as in the presented case, radiographic findings could mimic a bone malignancy . HoweverIn conclusion, we describe a rare case of a 12-year-old boy with a transverse stress fracture on a pre-existing NOF. With this case, we aim to point out that such a constellation in a child with local pain, together with discordant radiographic features, may be mistaken for a bone sarcoma. Careful history-taking and precise analysis of radiographic data could help avoid misdiagnoses. Even the mildest radiographic findings suggestive of a malignancy should be scrutinized and never overlooked. In case a satisfactory explanation for each of them cannot be found, additional imaging studies are warranted. Insufficiency fractures need serial radiographs to establish fracture healing.


Diseases of the uterus and adnexa uteri should be considered when evaluating female patients with lower abdominal pain. Diseases caused by defects in the supporting ligaments of the female reproductive system should also be considered. Diseases of the uterus and adnexa uteri should be considered when evaluating female patients with lower abdominal pain. Diseases caused by defects in the supporting ligaments of the female reproductive system should also be considered. An emergency laparotomy revealed an internal hernia of the sigmoid colon through a left mesosalpinx.Diseases of the uterus and adnexa uteri are generally considered among the diagnostic possibilities when evaluating female patients with acute lower abdominal pain in emergency department (ED) settings. However, diseases caused by defects in the supporting ligaments of the female reproductive system may also cause lower abdominal pain. Here, we present a case of a woman with an internal hernia of the sigmoid colon that occurred through a mesosalpinx defect.2A 38‐year‐old woman presented to our hospital with continuous acute abdominal pain, rated as a severity of 10/10 on the numerical rating scale. She reported several episodes of loose stools, but no hematochezia, nausea, or vomiting. The pain was located centrally in the lower abdomen and had not moved or spread. The patient reported an irregular menstrual cycle. Her last menstrual period had started 10 days prior and had lasted for 7 days. She did not experience atypical genital bleeding or hematuria with back pain. She had a medical history of caesarean section and untreated uterine fibroids, but denied any current gynecological diseases or inflammatory bowel diseases.2, 100% with room air; body temperature, 37.2°C; and Glasgow Coma Scale score, E4V5M6. A physical examination revealed tenderness in her middle lower abdomen without muscular guarding. Laboratory investigations revealed a white blood cell count of 7200/mm3. A pregnancy test was negative. Abdominal ultrasonography (US) did not indicate pyelectasis or a mass lesion in her lower abdomen. Abdominal computed tomography (CT) performed to evaluate the cause of the abdominal pain revealed stenosis of the sigmoid colon with dilation on the oral side, with no abdominal free air, significant ascites, or colon diverticula (Figure Her vital signs were as follows: blood pressure, 120/69 mm Hg; heart rate, 100 beats/min; respiratory rate, 19 breaths/min; SpOAn emergency laparotomy was performed, which revealed strangulation of the sigmoid colon by a fallopian tube, ovarian suspensory ligament, and ovarian ligament. The patient was diagnosed with an internal hernia of the sigmoid colon through a defect in the left mesosalpinx. The strangulated colon was edematous, and there was a small amount of ascites in the abdominal cavity. The herniated colon was not necrotic and was released from ligamental strangulation. Her postoperative recovery was uneventful, and she was discharged on the fourth day after surgery.3Evaluations of patients with acute abdominal pain in the ED generally consider common diseases and acute abdominal or vascular diseases, such as aortic dissection or mesenteric ischemia.Internal hernias involve a protrusion of the viscera through the peritoneum or mesentery and into a compartment of the abdominal cavity.We have identified five previous case reports describing internal hernias of the intestine through the mesosalpinx Table .6, 7, 8,In four of the five reported cases, surgery was performed because abdominal radiography suggested an intestinal obstruction. In one case, small bowel resection was performed because of mesenteric ischemia. In our case, CT enabled a diagnosis of mesenteric obstruction, and colonoscopy revealed impaired blood flow to the intestine. These findings enabled us to perform an emergency laparotomy.4Diseases of the uterus and adnexa uteri should be considered when evaluating female patients with lower abdominal pain. In such cases, diseases caused by defects in the supporting ligaments of the female reproductive system should also be considered, especially since such conditions need emergency laparotomy.None.TH: is the first author, managed the patient in the emergency department, and drafted the manuscript. SU: reviewed and edited the manuscript, prepared the manuscript for the submission, and communicated with editors. AK: reviewed and edited the manuscript and supervised the writing and submitting process. All authors discussed and approved the submitted manuscript.


We report two resected cases of solitary fibrous tumors (SFT) that were accidentally found in the pelvic cavity.Case 1 was a 54-year-old male. A colonoscopy for the examination of intestinal polyps revealed an extramural tumor in the right anterior wall of the low rectum. A preoperative MRI showed a well-demarcated T1 low and T2 mixed intensity extramural tumor (53 × 36 mm) located right lateral to the low rectum and behind the seminal vesicle. Laparoscopic surgery was successful for tumor extirpation. Immunohistochemical examination of the specimen revealed STAT6 (+) and CD34 (+) cells, a Ki67 positivity of 7–8%, a mitotic index of 4–5/50 HPF, and a diagnosis of SFT. There was no recurrence 29 months after surgery. Using RT-PCR and sequencing, we detected the NAB2-STAT6 fusion gene but the locus of genomic inversion was not detected. Case 2 was a 43-year-old male that received conservative treatment for appendicitis. A CT scan accidentally revealed a tumor of 40 mm of length in the left obturator area. A MRI revealed a well-demarcated T1 and T2 high intensity tumor. The patient underwent surgical biopsy. Immunohistochemical examination of the biopsy revealed STAT6 (+) and CD34 (+) cells, Ki67 positive cells < 1%, and a diagnosis of SFT. We could not detect the NAB2-STAT6 fusion gene in the extirpated tumor.Two cases of pelvic SFT were diagnosed by immunohistochemical examination, RT-PCR and sequencing and successfully resected by laparoscopic surgery. A total of 43 cases of pelvic SFT were reported in the literature in Japan. However, in only 5 cases reported STAT6 protein overexpression by immunohistochemical staining, the product of the NAB2-STAT6 fusion gene, a driver mutation of the SFT [Solitary fibrous tumors (SFT)  are mese2Case 1: A 54-year-old male underwent a colonoscopy for the examination of colon polyps that did not detect any tumor lesions but revealed an extramural compression in the right anterior to lateral wall of the lower rectum. A CT scan revealed a well-demarcated encapsulated tumor (maximum length 60 mm) with contrast enhancement at the right anterior to lateral side of the rectum. A preoperative MRI showed a well-demarcated T1 low, T2 mixed intensity extramural tumor behind the seminal vesicle located between the right lateral wall of the low rectum and the right lateral pelvic wall .Fig. 1Pr2.1The preoperative diagnosis was gastrointestinal stromal tumor (GIST). Laparoscopic tumor resection was performed in March 2017. Based on the MRI findings, the peritoneum was incised outside the vesicohypogastric fascia to expose the internal obturator muscle following dissection between the left pelvic fascia and mesorectal fascia. Secondly, the tumor in the vesicohypogastric fascia was isolated from both sides. Finally, the tumor was removed from the vesicohypogastric fascia and levator ani muscles. The surgery lasted 388 min and the total amount of blood lost was 502 mL.2.2Macroscopic findings revealed a smoothly lobulated mass (53 × 36 mm) encapsulated by a thin, translucent membrane. In addition, the cut surface appeared grey white to tan with a whorled pattern. Microscopic findings revealed the tumor consisted of spindle cell tumors complicated in bundles, and necrotic tissue with vascular and collagen fiber growth. Except for the hemostatic cauterization of the tumor vessels, the capsule structure was pathologically intact.The tumor was STAT6 (+), CD34 (+), DOG1 (−), cKIT (−), S100 focal (+), and Desmin (−) by immunohistochemistry. The NAB2-STAT6 fusion gene was also confirmed. Ki67 positive cells represented 7–8% and Fission image 4–5/50 HPF resulting in a diagnosis of malignant SFT , Fig. 4.Case 2: A 43-year-old male was referred to our hospital complaining of pain in the right lower abdominal cavity and was diagnosed with appendicitis. A preoperative plain CT scan revealed a well-demarcated tumor (maximum length 40 mm) at the left obtulator fossa in contact with the left lateral wall of the rectum. He underwent laparoscopic appendectomy and a tumor biopsy via the retroperitoneal route. Histopathological examination of the biopsy specimen revealed it consisted of spindle tumor cells STAT6 (+), CD34 (+), DOG1 (−), cKIT (−), S100 (−), Desmin (−), CDK4 (−), PAX8 (−), and Ki67 positivity < 1% by immunohistochemistry. The tumor was diagnosed as SFT. A preoperative MRI after intraoperative biopsy revealed the tumor was a well-demarcated T1 and T2 low intensity tumor. In FDG-PET, an accumulation of SUVmax 2.3 corresponding to a mass was observed .Fig. 5Pr2.3Based on the MRI findings, the Retzius cavity was dissected followed by dissection between the left side mesorectal fascia and pelvic plexus. The peripheral side of the neurovascular bundle was divided at the left lateral side of the prostate. The peritoneum was incised outside the left vesicohypogastric fascia to expose the internal obturator muscle. The tumor in the vesicohypogastric fascia was then isolated from both sides. Finally, the proximal side of the branches of the internal iliac vessels and the pelvic plexus were divided and the tumor removed together with the vesicohypogastric fascia. The surgery lasted 228 min and the total amount of blood loss was 168 mL.2.4Macroscopic findings revealed a smoothly lobulated mass (maximum length 40 mm) encapsulated by a thin membrane that was not detected by microscopic examination. The cut surface was grey white to tan with a whorled pattern. Microscopic examination revealed spindle tumor cells. The immunohistochemical findings were the same as the results of the biopsy sample , Fig. 8.3SFTare rare neoplasias of mesenchymal origin with low grade malignant potential, deriving from a wide range of anatomic sites. Gold et al. reported that 16% of their series of SFT derived from the pelvic cavity . Though To the best of our knowledge, including our own cases, 45 cases of pelvic SFT have been reported in Japan of which 27 derived from the surgical department followed by 10 reports from urology, 3 from radiology and one from gynecology. There were no gender differences in the incidence with a median age of 58 (19–83) years. The median size of the tumor was 10 cm (0.85–29.0). Most of the cases were reported as large tumors with compression symptoms such as abdominal distension, frequent urination, and dysuria. Surgery was the only curative treatment, sometimes requiring a trans-sacral approach in addition to the open abdominal approach . In a reen-bloc R0 resection is necessary also in neurosurgery, the rate of R0 resection varies depending on the tumor location [SFT are not easy to diagnose preoperatively using imaging. Although MRI was often reported as T1 low and T2 high intensity, it was not always a characteristic finding of SFT. In FDG-PET, some reports revealed no uptake in the tumor. Case 2 showed a slight accumulation, but its significance is unknown. As a consequence, 17 cases were diagnosed by preoperative biopsy. In case 2, STAT6 immunohistochemical staining of the biopsy sample revealed the SFT. Most cases of SFTs behave as benign tumors, but 15–20% of them experience distant metastasis as well as local progression. Although en-bloc resection of the tumors without injury. For the tumor located in the obturator fossa, the technique of laparoscopic lateral pelvic lymph node dissection that was widely accepted in high volume centers of colorectal surgery in Japan was available.In the cases presented here, tumors were detected below the peritoneal reflection involving the pelvic plexus and vesicohypogastric fascia with a relatively small size. As the resection of the pelvic plexus and vesicohypogastric fascia in combination with the tumor required clear anatomical recognition in the deep pelvic cavity throughout the operation, laparoscopic surgery was suitable for Recent studies revealed that the NAB2-STAT6 fusion gene was a driver mutation of SFT. The expression of NAB2-STAT6 fusion proteins had the early growth response (EGR)-binding domain of NAB2 fused to the activation domain of STAT6. Based on this review, only 5 cases were examined by STAT6 immunostaining (only one report described the presence of NAB2-STAT6 fusion gene) . Based o4In addition to this report, there are only 7 cases of pelvic SFT showing STAT6 immunohistochemical staining. It is expected that accumulation of data on NAB2-STAT6 fusion gene will elucidate the pathophysiology of pelvic SFT.No supportive foundations. No conflict interest has been declared by Yuki Matsui, Madoka Hamada, Fusao Sumiyama, Toshinori Kobayashi,Yuki Matsumi, Hisanori Miki, Mitsuaki Ishida, Hiroaki Kurokawa, Mitsugu Sekimoto, Yoko Sekita-Hatakeyama, Kinta Hatakeyama, Chiho Ohbayashi.No supportive foundations. No conflict interest has been declared by Yuki Matsui, Madoka Hamada, Fusao Sumiyama, Toshinori Kobayashi,Yuki Matsumi, Hisanori Miki, Mitsuaki Ishida, Hiroaki Kurokawa, Mitsugu Sekimoto, Yoko Sekita-Hatakeyama, Kinta Hatakeyama, Chiho Ohbayashi.http://www.kmu.ac.jp/hirakata/hospital/2671t8000001356c.html).This study was approved by the Hospital Ethics Committee of Kansai Medical University .This study was approved by the Hospital Ethics Committee of Kansai Medical University (reference number #2019230: All authors were involved in preparation of this manuscript.Not commissioned, externally peer-reviewed journal.


Despite its first identification in 1885, intraluminal duodenal diverticulum remains a rare entity and only a few case reports are found in the literature. Its diagnosis is almost always delayed due to the lack of specific symptoms and to the very vague presentation consisting of mild epigastric discomfort. However, with the aid of new diagnostic modalities and imaging, it has become easier to diagnose this entity when its symptoms persist. Finally, it can remain undiagnosed in asymptomatic patients. We present a case of 21-year-old female patient with a negative medical and surgical history. She presented to the gastroenterology clinic for chronic epigastric pain for several months, associated with postprandial episodes of nausea and vomiting. Her physical examination was insignificant. She was prescribed proton pump inhibitors and prokinetics.However, her symptoms didn't improve much, so a diagnostic upper esophagogastroduodenoscopy was done and showed normal mucosa but with external compression on the second part of the duodenum . ComputThe patient was presented with her options and after a surgical consult, and due to her intractable symptoms, it was decided to go with surgical treatment. Written consent was taken from the patient. She was prepared for the operating room.Intraoperatively, the duodenum was first completely exposed. Then, an intraluminal mass was palpated at the level of D2. Through a longitudinal duodenotomy, the intraluminal diverticulum was identified. The diverticulum was identified 20-mm proximal to the papilla. The papilla was intubated to avoid injury during traction of the mucosa. Excision of the diverticulum was then performed and the mucosa was approximated using absorbable sutures.The postoperative course was unremarkable and the patient was discharged on day 6 after regaining bowel movement and tolerating oral intake. During her follow-up in the outpatient department, a week after her discharge, she reported complete resolution of her preoperative debilitating symptoms of pain and nausea.By definition, a diverticulum is a sac-like protrusion that can occur through gastrointestinal (GI) tract. Diverticula of the small bowel are very uncommon and tend to be asymptomatic. They are usually found incidentally on imaging. Approximately 2 to 5% of patients undergoing upper GI barium studies and 7% of patients undergoing endoscopic retrograde cholangiopancreatography (ERCP) have been reported to have duodenal diverticula. Diverticula are found in most cases in the second portion of the duodenum.According to the shape and the etiology of the diverticula, they are classified as extraluminal duodenal diverticulum (EDD) or IDD.EDD are acquired and consist of an outpouching of mucosal and submucosal layers that herniate through a muscular defect in the bowel wall. The precise mechanism for EDD development remains unknown. However, some theories suggest the presence of a low resistance area in the bowel wall, which may have been created by the passage of the biliary duct, pancreatic duct, or blood vessels through duodenal wall. Another theory proposes congenital causes like the absence of an adequate muscle coat or heterotrophic pancreatic tissue.IDD or “windsock” diverticulum is a protrusion of the mucosa into the lumen of the duodenum. It is a rare congenital abnormality that is probably due to an incomplete recanalization of the foregut lumen.IDD is actually becoming more and more common in adults due to the increased use of radiologic and endoscopic investigations. The reported frequency is as high as 22% at time of autopsy. Note that, less than 10% of duodenal diverticula are actually symptomatic with less than 1% requiring definitive treatment.The median age of diagnosis is between 30 and 39 years, but it can also be found in pediatric ages.Diagnosis of IDD is difficult upon presentation. Imaging is always necessary to confirm the diagnosis. Around half of them (55%) are seen on upper GI contrast studies. Nevertheless, CT remains more sensitive because the endoscopy can report normal mucosa and normal duodenal appearance in cases of IDD; whereas CT scan can easily identify a globular out-pouching in the duodenum.Surgical treatment of asymptomatic duodenal diverticulum is not indicated because there is no risk of malignant transformation. Intervention is only required for symptomatic patients who are nonresponsive to medical treatment, or who develop complications such as bowel obstruction, GI bleeding, or pancreatitis. Historically, gastrojejunostomy was performed for gastric outlet obstruction, prior to the identification of the IDD. Nowadays, excision of the diverticulum has become the treatment of choice. The excision can be performed endoscopically (first performed in 1979), or surgically via an antimesenteric duodenotomy after identification of the ampulla of Vater.


Due to its rarity, little is known about the clinical presentations and responses to systemic chemotherapies in advanced and/or metastatic cases of paratesticular liposarcoma.Here, we report the case of a 75-year-old Japanese man with giant paratesticular liposarcoma. Imaging studies revealed a 26 cm tumor in his right scrotum and lung metastases at presentation. He underwent radical orchiectomy followed by systemic chemotherapies. Pathological findings of the resected primary tumor confirmed a dedifferentiated liposarcoma. He then started chemotherapy treatment with gemcitabine plus docetaxel. His disease status was stable for 1 year. Eribulin was used for second-line chemotherapy. He had a relapse at 5 months after eliburin and died at 22 months after diagnosis.Early diagnosis and curative radical surgery are important for treatment of paratesticular liposarcoma. However, a giant paratesticular liposarcoma could cause metastases, and systemic chemotherapy may be helpful for prolonging survival in patients with metastatic paratesticular liposarcoma. Urological sarcomas are rare tumors accounting for 2.7% of all soft tissue sarcomas across organs in adult patients . Parates2 day 1) plus docetaxel  chemotherapy every 2 weeks. The metastasis in his left lung was set as a target lesion by chest radiography. The time course of the imaging studies is shown in Fig. 2, day 1, 8, every 3 weeks) chemotherapy. Despite the progression of the lung metastases, his symptoms remained stable. However, at 4.5 months after second-line chemotherapy, chest radiography showed progression of metastatic nodules and the presence of pleural effusion. In all, he underwent chemotherapy for 18 months and died at 22 months after diagnosis. An autopsy was not performed.A 75-year-old Japanese man complained of a swollen right scrotum, which had grown slowly for 1 year. A huge elastic hard mass was palpable in his right scrotum. The surface of the skin showed no redness, rash, ulcers, or other skin abnormalities. He had neither pain nor obstruction of urination. He had worked as a farmer for a long time. He had a smoking history (20 pack-year) but did not consume alcohol. There was no history of disease or surgery. There was no specific family history, including of cancer. Physical and neurological examinations were normal, except for a 26 cm tumor in his right scrotum and right inguinal lymphadenopathy 3 cm). His blood pressure was 146/80, heart rate 86, and temperature 36.8 degrees. Laboratory findings are shown in Table  cm. His We described a case of a patient with giant paratesticular liposarcoma initially presenting with pulmonary metastases, and the treatment outcome along with serial chemotherapies. Such a huge tumor is extremely rare, and little information about the usefulness of chemotherapy exists about paratesticular liposarcoma.Malignant liposarcoma is classified into the following subtypes: dedifferentiated, myxoid/round cell, and pleomorphic liposarcoma . Like otet al. examined the differential sensitivity of liposarcoma subtypes to chemotherapy [There were no metastatic lesions in organs other than lungs, and systemic chemotherapy was initiated in the present case. The response rates of chemotherapies, such as doxorubicin, ifosfamide alone, or in combination, are generally low and the response rates to these agents also vary according to histological types in soft tissue sarcomas . Jones eotherapy . Myxoid otherapy  and the otherapy , gemcitaotherapy . Althougotherapy , informaEribulin, a microtubule growth inhibitor, was highly efficacious in patients with advanced liposarcoma . HoweverIn the SEER data, median survival time in registered patients with advanced liposarcomas was 21.1 months . The surWe described a case of giant paratesticular liposarcoma initially presenting with pulmonary metastases, along with the treatment outcome. Although systemic chemotherapy may be helpful for prolonging survival, further studies regarding therapeutic strategies for inoperable and metastatic paratesticular liposarcomas are warranted.


Background: Ear click is a rare type of objective tinnitus, classically described with associated palatal tremor/myoclonus (PT). Case report: A 15-year-old boy reported a constant bilateral ear clicking for 4 years, that could be stopped at will for a few seconds. Clinically, the ear clicks were audible without visible eardrum or palatal movement, and could be entrained by the examiner. Brain MRI was normal. Discussion: We propose to classify this as isolated ear clicks with partial voluntary control, putting it into context with other subcategories of “essential” or “isolated” PT. One form of objective tinnitus  is referred to as an “ear click”. Ear click can be due to tremor or myoclonus involving the muscles attached to or surrounding the Eustachian tube , in which case it is classically described with associated palatal movement. We describe a case of objective clicking tinnitus without palatal tremor/myoclonus (PT) in which the ear clicks had semi-volitional characteristics.A healthy 15-year-old boy reported a 4-year history of persistent spontaneous tinnitus in both ears. The tinnitus consisted of constant “clicks” at an irregular frequency, heard on both sides, independent of heart rate, present throughout the day but not during sleep. Clicks were not associated with other facial, cranial or body movements. There was no history of head trauma. No specific psychological trigger was identified. Neither the patient nor his family had any history of tics. The patient denied any urge or relief produced by the movements, and there was no waxing or waning. The patient reported the ability to willingly stop the tinnitus for a few seconds, especially by performing Valsalva maneuver. He denied any hearing or balance problem and had no history of neurological or otological disorder.On clinical exam, audible irregular “clicks” were perceived on both sides of the patient’s head. Otoscopy revealed normal tympanic membranes without any visible movement . PosteriPure tone audiometry, tympanometry and stapedial reflexes were normal. Full brain MRI with thin cuts was normal. A trial with Clonazepam 0,5 mg bid for a few weeks didn’t change the symptomatology and was stopped by the patient.Objective tinnitus can be caused by temporomandibular joint mechanical disorders, vascular abnormalities, patulous Eustachian tubes, middle-ear myoclonus (MEM), or associated with PT. Symptomatology can be distressful, requiring psychological support. Management options include medical treatments such as benzodiazepines and anticonvulsants, botulinum toxin injections and middle-ear muscles tendon section in selected cases .3) in MEM, and contraction of peritubal muscles  VII) or tensor tympani (CN V muscles . MEM ref3) in essential or isolated PT, or the levator veli palatini (CN IX and X) in the rare cases of symptomatic PT with ear clicks [In case of ear clicks due to peritubal muscle contractions, changes in apposition of Eustachian tube walls by repetitive openings is believed to produce the sound . These cr clicks 8. Another clicks . This laessential” nosology in this isolated ear click case. Essential/isolated [11By comparison with classical PT, the absence of both an MRI lesion and associated neurological signs would point towards an “isolated  (i.e. noisolated 1112. In


Actinomyces is a rare cause of prostatic abscess. Most reported cases of abdominal and pelvic actinomycosis are a consequence of invasive procedures or abdominal infections. In this report, we present a previously healthy 53-year-old man with inguinal pain, fever and dysuria who was found to have a prostate abscess, secondary to Actinomyces turicensis and Peptostreptococcus, which was removed via transurethral resection of the prostate. This case was complicated by Peptostreptococcus bacteremia and a facial abscess. The patient was treated with antibiotics and ultimately made a full recovery. Prostatic abscesses are uncommon but potentially serious disorders. They may occur as sequelae of bacterial prostatitis or urinary reflux, or may result from hematogenous spread from other organs . They arA 53-year-old man presented with a one-week history of left inguinal pain, subjective fever and chills, and two episodes of vomiting. Two days prior to presentation, he also developed incomplete bladder emptying and dysuria. The patient had no significant past medical or surgical history. There was no history of prior lower urinary tract symptoms, genitourinary instrumentation or catheterization, or of renal stones. There was a history of a gunshot wound to right posterior chest wall 35 years previously, with a retained bullet.9/L (neutrophil count: 25.5 × 109/L). He was unable to tolerate a digital rectal exam due to pain. Urinalysis showed large leukocyte esterase, blood and moderate bacteria. Bladder sonography revealed a postvoid residual of 60 cc. CT of the abdomen and pelvis revealed a large 6 x 2.7 cm prostate abscess tracking into the seminal vesicles bilaterally and an incidental 5 x 2.5 cm left inguinal hernia  and transfer to the medical ICU. He stabilized two days later; he was successfully extubated and transferred to the urology service.A repeat CT was performed which showed resolution of the prostatic abscess and a new left inguinal cord abscess Figures , 1d, whiActinomyces turicensis and Peptostreptococcus are especially rare causes of prostatic abscess. Actinomyces is a genus that consists of several species of gram-positive anaerobic bacilli . These sOver the past few decades, several new species of Actinomyces have been discovered. Actinomyces turicensis was first described in 1995 .Actinomyces turicensis infections may occur with concomitant organisms including fellow anaerobes, such as Peptostreptococcus . As gramThe role of anaerobes in precipitating prostatitis, and thus prostatic abscess, is likely underestimated, as they are not routinely cultured in urine . This isThere are no standard guidelines for the management of prostatic abscess given that it is a rare condition; however, in general, treatment should include parenteral broad-spectrum antibiotic administration and drainage of the abscess . AbdelmoIn this report, we describe a prostatic abscess secondary to Actinomyces and Peptostreptococcus complicated by sepsis in a previously healthy middle-aged man with no identifiable predisposing factors. The original source of the abscess is unknown, but the incidental hernia discovered on initial imaging and development of an inguinal cord abscess possibly implicates the gastrointestinal tract. Additionally, hematogenous spread of Peptostreptococcus to the face may explain subsequent formation of a facial abscess. The patient made a recovery to near baseline after transurethral resection of the prostate, hernia surgery, incision and drainage of the facial abscess, and antibiotic therapy.


Post-appendectomy mucus-filled lesions, arising from appendiceal remnant tissue, are an exclusively rare clinical entity. We report a rare case of a mucus-filled lesion rising from a distal viable remnant appendiceal tip as a late complication of appendectomy.A 48-year-old man presented with a 2-week history of right buttock and right lower quadrant abdominal pain. He had previously undergone appendectomy 23 years ago. Radiological examination showed a cystic lesion lateral to the colon with a prominent tubular structure. According to the past operative note, the appendiceal tip was lost accidentally. Surgical resection was performed. Pathologic examination revealed a mucus-filled lesion lateral to the cecum, with no communication between them. There was no evidence of malignancy. The prominent structure had muscular layer, suggestive of distal appendiceal remnant.Surgical resection of appendiceal mucus-filled lesion is recommended, because there are no reliable criteria to exclude malignant lesions. Although appendiceal mucus-filled lesion is rare, it is extremely rare in patients with previous appendectomy. In our case, the mucinous material secreted from the remnant appendix might be enclosed by the surrounding tissue.In patient with previous appendectomy who show a cystic mass close to the cecum, the possibility of lesion derived from the appendiceal stump or distal appendiceal remnant should be considered. In such cases, it is important to review the medical records for a definitive diagnosis. Complete appendectomy, without leaving appendiceal tissue, remains mandatory to prevent the development of late complications of mucus-filled lesions. Preoperative diagnosis is important to prevent intraoperative perforation, which may result in pseudomyxoma peritonei . AppendiWe present a rare case of mucus-filled lesion derived from a viable leftover appendiceal tip as a late complication of open appendectomy.The work has been in line with the SCARE criteria 72An active 48-year-old man presented with a 2-week history of right buttock and sudden right lower quadrant (RLQ) abdominal pain. He had normal appetite and bowel habits. His past medical history was unremarkable except for an acute appendicitis and open appendectomy 23 years prior to this visit. His body weight, height, and body mass index were 95.2 kg, 178.0 cm and 30.0, respectively. He was a hotel worker and had a cigarette smoking history of one pack a day. There was no family history of malignancies.At presentation, his vital signs were stable. Physical examination was remarkable for a soft, nondistended abdomen with tenderness in the right lateral to lower abdomen with past appendectomy scar. There was no rebound tenderness or guarding. An emergent condition was suspected, including diverticulitis, ileitis, and enteritis.Serum tumor markers and cancer antigen 19–9 (CA19-9 2.2 U/mL) were within normal range; however, carcinoembryonic antigen  levels were elevated.Abdominal contrast-enhanced computed tomography showed a well-defined hypodense (15–20 Hounsfield units) cystic mass lesion measuring approximately 7.0 × 12.0 cm lateral to the cecum, which appeared not to be communicating . The lesMagnetic resonance imaging showed a well-circumscribed lesion that was markedly hypointense and hyperintense on T1- and T2-weighted images, respectively .Colonoscopic findings were normal; however, the appendiceal orifice could not be identified.Schematic illustration of the expected ileocecal lesion is shown .Fig. 2ScPast operative note revealed that his past open appendectomy 23 years prior had several issues. The spinal anesthetic effect was not stable, and skin incision was done closer to the midline than usual. The appendix was found to have a long body with the distal tip strongly adhered to the retroperitoneum; therefore, retrograde appendectomy was performed. During tissue peeling, the appendix accidentally detached, and the tip was lost . DespiteHistopathologic examination revealed phlegmonous appendicitis without any malignancy. Postoperatively, he complained of RLQ abdominal pain, and computed tomography (CT) showed inflammation of the periphery of ileocecum at 2 weeks after surgery. His symptoms resolved transiently after antibiotic therapy.Considering the possibility of infectious diseases, the patient received empirically intravenous antibiotics (piperacillin/tazobactam); however, his abdominal pain and inflammatory markers did not improve.Conducting elective exploratory laparotomy with preceding ureteral stent placement found a well-encapsulated mass, adherent to the surrounding tissues lateral to the cecum, which had a prominent structure similar to a distal appendiceal remnant. The wall was mildly inflamed, but there was no evidence of perforation or abscess formation. There was no lymph node enlargement, and the mass was not easily mobilized from adjacent structures. Therefore, en bloc ileocecal resection with sufficient margins and systematic lymph node dissection were performed.Gross pathologic examination revealed a distended mucus-filled lesion lateral to the cecum without communicating with the lumen . The mucThe patient was diagnosed with a mucus-filled lesion rose from the distal viable remnant tip of the appendix . PeriopeThe patient’s postoperative recovery was uneventful. He was symptom-free and maintained a normal CEA level (2.2 ng/mL) during the 4-month follow-up period.3Appendiceal mucus-filled lesion, called mucocele, is a rare clinical entity first described in 1842 . In 2012Clinical presentations of appendiceal mucocele are nonspecific, including abdominal pain with appendicitis, nausea and vomiting, weight loss, palpable mass, changes in bowel habits, or lower gastrointestinal bleeding;  for thesPreoperative diagnosis is not only important to prevent perforation, which may result in pseudomyxoma peritonei , but alsSurgical resection of all appendiceal mucinous lesions is recommended for both diagnostic and therapeutic purposes, because there are no reliable criteria to exclude malignant lesions . An algoDuring spontaneous perforation, leakage from the cyst, enlarged mesenteric lymph node, or positive cytology strongly suggestive of malignancy, right hemicolectomy should always be performed to achieve complete cure ,13.18The RLQ abdominal pain in adults who have undergone appendectomy represents a diagnostic challenge. The WISS study confirmed acute appendicitis as the most frequent cause of intra-abdominal infection,  and appeStump appendicitis (SA) is a rare case of inflammation of remnant appendiceal tissues due to the incomplete removal of the appendix ,20, AlthNine cases of post-appendectomy mucus-filled lesion (PAML) were reported . As prev6 in which the lesions were also derived from the distal viable remnant tip, our case had a non-epithelial wall. Similar to SA, the mechanism of PAML formation is likely due to incomplete removal of the appendix [Compared to Johnson et al.’s study, appendix . Here, tSince an unusual case of simple mucocele with elevated serum CEA had been reported,  consider4Ultimately, in patient with previous appendectomy who showed a cystic mass close to the cecum, not connected to the lumen, one should take the possibility of PAML derived from appendiceal stump or distal viable remnant appendiceal tip into consideration.It is also important to review the histology of the appendix and previous operative notes for definitive diagnosis. The surgical treatment of acute appendicitis has undergone a paradigm shift from open to laparoscopic surgery. However, complete appendectomy, without leaving appendiceal tissue, remains mandatory to prevent complications of mucus-filled lesions.None declared.NoneEthical approval has been exempted by my institution.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.HT wrote the draft of the manuscript. SH revised the article. HT and SH performed perioperative care and performed the operation. MK is the pathologist. All authors participated in the management of the patients in this case report.Not applicable.Shinichi Hashimoto.Not commissioned, externally peer-reviewed.


A 61 years old male patient presented to the Accident and Emergency (AE) Department reporting bleeding during defecation, difficulty and low pain while walking and sitting. On physical examination we saw an exophytic cauliflower like, ulcerated tumor mass measuring 20 x 14 cm that covered the whole perineum and a similar satellite lesion 3 x 4 cm, with malodorousness. The mass was there for at least three years, progressively growing.


Idiopathic mandibular condylar resorption is a rare condition in which the mandibular condyle of the temporomandibular joint (TMJ) becomes resorbed and thus reduces in size and volume. This leads to TMJ dysfunction that commonly requires surgical correction; however, more conservative interventions can also be utilized. We present a case of idiopathic mandibular condyle resorption in a 17-year-old female presenting with TMJ pain and clicking with mastication. A definitive diagnosis of this condition ultimately requires imaging studies, a reliable option being magnetic resonance imaging (MRI), which will reveal erosion of the mandibular condylar process  with diminished mass and volume leading to the known sequelae of symptoms. Mandibular condylar resorption is a rare condition in which the condylar process of the mandible is progressively resorbed and thus reduces in size over a period of time . This phIf the condylar process undergoes erosion at the TMJ, the joint naturally retracts posteriorly, resulting in mandibular retrognathia, malocclusion of the teeth, and open bite deformities -4. PatieA 17-year-old female presented to her primary care physician with recurrent jaw pain and clicking upon TMJ manipulation. She had no significant past medical history at the time of presentation and did not experience any prior trauma or any surgeries in the past. She denied any other joint pain or systemic symptoms.Physical examination was unremarkable, except for induced pain and a clicking sound appreciated bilaterally upon TMJ movement. Maxillofacial computed tomography (CT) was ordered Figures -4 which Whether the bone loss that occurs in condylar resorption is due to hormonal influences, inflammation, or overwhelming compressional forces, there is a common resorption pathway involved 1). This pathway involves cytokines, e.g., tumor necrosis factor-alpha (TNF-α), interleukin-6 (IL-6), or receptor activator of nuclear factor kappa-B ligand (RANKL), that activate osteoblasts in the associated bone [. This paEstrogen is known to play a role in bone and cartilage metabolism . It has Given our patient’s lack of significant past medical history, trauma, or surgeries, we can assume she experienced primary (or idiopathic) mandibular condylar resorption. Her age and gender also help to further solidify this assumption as the hormonal changes seen in this demographic are thought to contribute to the idiopathic category of this condition. Idiopathic condylar resorption is ultimately a diagnosis of exclusion once other potential (secondary) causes have been ruled out .Imaging studies are imperative in arriving at a definitive diagnosis of condylar resorption as the remainder of the diagnostic criteria are based on clinical findings and patient history. Magnetic resonance imaging (MRI) is a reliable imaging modality to identify changes related to condylar resorption, and findings will be remarkable for a notable decrease in condylar size and volume, which is often bilateral -5. AnterThere has been minimal evidentiary support for the use of pharmacologic agents in the treatment of condylar resorption . ManagemIdiopathic mandibular condylar resorption is a rare condition affecting predominantly females within the pubertal age range. Hormonal changes are thought to play a role in the pathogenesis of idiopathic condylar resorption, explaining the most commonly affected demographic identified in clinical practice. Although surgical correction is the mainstay of treatment, a deeper understanding is required before an etiologic-based treatment can be established.


A Male child of eighteen months old was admitted in the pediatric medical emergency department for ingestion of foreign body while he was playing with his seven years old sister. He was not presenting any symptoms. The clinical examination was normal: he was conscious, calm, with no respiratory distress. The inspection of oropharynx did not find any abrasions or lacerations and the abdomen examination was normal. A thoraco abdominal X-ray showed an image of a knife measuring 12cm between the lower third of the esophagus and the stomach. A second X-ray with the undressed child confirmed the diagnosis (A). A second anamnesis made the sister confess that they were watching a television show of sword swallowers when her brother ingested the knife. The patient was admitted in the operating room to extract the sharp object. The surgical removal did not find any lesions of the gastrointestinal tract and was successful (B).


A prompt diagnosis is mandatory to avoid fatal complications in case of pulmonary artery pseudoaneurysm. We report a rare case of post‐operative pulmonary artery pseudoaneurysm. In such instance a prompt diagnosis and adequate treatment are essential to avoid catastrophic complications. Moraxella catarrhalis was isolated from sputum and tissue cultures. Post‐operative pulmonary artery pseudoaneurysm is a rare event described in only few case reports [A 68‐year‐old woman was admitted to our unit for treatment of lung adenocarcinoma and scheduled for thoracoscopic left upper lobectomy. She had an uneventful hospital stay and was discharged on the fifth post‐operative day. After 24 h, the patient experienced a high fever and she was admitted to the hospital. Post‐operative chest computed tomography (CT) revealed a lung abscess in the left lower lobe Fig. . Since t reports , 2. In sAppropriate written informed consent was obtained for publication of this case report and accompanying images.


Malignant fibrous histiocytoma (MFH) is an undifferentiated high-grade pleomorphic sarcoma and is considered the most common primary soft tissue sarcoma in adults. MFH is known to arise in the trunk, extremities and retroperitoneum although it can arise anywhere in the body.MFH of the skin is uncommon and even less frequent is the involvement of the scalp, especially with skull invasion. Most of the MFH cases present as a gradually growing lesion over a period of one to two years and is associated with ulceration and hemorrhage in most of the cases. Treatment of MFH is usually surgical resection.We present a case of MFH in an 85-year-old gentleman that was invading the skull which required a multidisciplinary surgical treatment for resection and microvascular free flap reconstruction. Malignant fibrous histiocytoma (MFH) is a pleomorphic sarcoma, which is considered the most common primary soft tissue sarcoma in adults -5. MFH iAn 85-year-old gentleman with a history of basal cell carcinoma of the scalp seven years prior, which had been removed using Mohs chemosurgery, presented to his dermatologist with a complaint of a nodular indurated ulcerative lesion on the left parietal aspect of his scalp. The lesion had been present for several months, but the patient became concerned one month prior to presentation after his wife noticed bleeding and drainage of serous fluid from the lesion. The patient denied any other associated symptoms including headache, nausea, vision changes, numbness, tingling or weakness of either the upper or lower extremities.An attempt was made to excise the lesion in the dermatologist’s office utilizing a Mohs procedure. However, the procedure was aborted due to concerns that the lesion might be invading the underlying skull. The patient was subsequently referred to a plastic surgeon and neurosurgeon for further evaluation and care.Computerized tomography (CT) scan of the head Figure  and magnThe neurosurgery team performed a left craniectomy for an en bloc resection of the scalp and skull lesion followed by duraplasty and cranioplasty with titanium mesh. Subsequently, the plastic surgery team performed a microvascular free flap tissue reconstruction of the 7.5-cm-diameter circular scalp defect. A left radial forearm fasciocutaneous free flap was harvested and anastomosed to the superficial temporal vessels  with osseous invasion , lymph nodes 35%), bone (8%) and the liver 1%) 7]. The t5%, bone We reported a case of MFH of the scalp with invasion through the skull in an 85-year-old gentleman. CT scan of the head and MRI of the brain demonstrated erosion through both the outer and inner tables of the skull. Treatment of MFH required a multidisciplinary surgical treatment for resection and microvascular free flap reconstruction. Diagnosis of MFH is challenging, and it is important to differentiate MFH from other dermatologic pathologies, such as squamous cell carcinoma and malignant melanoma. Histological features and immunohistochemical markers are necessary for definitive diagnosis. Surgical excision is the definitive treatment. The excisional wound often does not permit primary closure and necessitates some form of complex reconstruction.


Central giant cell tumors (CGCTs) bones are uncommon benign bone tumors in the jaw.The authors report a pediatric patient who was diagnosed with an aggressive central giant cell cyst of the mandible and its management. The CGCT usually occurs in adolescents patients from 5 to 10 years of age. An 8-year-old female patient presented with progressive non-pulsatile swelling of the mandible. Intraoral examination revealed mobility of teeth, associated with gingival swelling.Common sites of giant cell tumors are the epiphysis of the long bones. Giant cell tumors accounted for 4–7% of all primary bone tumors. The mandible localization is exceptional, the main treatment for giant cell tumors is surgical excision and different surgical approaches had been reported in the literature thus depending on the size of the cyst, localization, and experience of the surgeon.Giant cyst tumor of the jaw is rare and less common in childhood. The clinical behavior is very diverse. The main treatment is surgical removal and follow-up to reveal recurrence. She was referred to our department's consultation for specialized care. No other personal or family history was raised during the patient interrogation. Clinical examination revealed large tender swelling without sensory disturbances and incompetent lips with a protruding lower lip. In the vertical direction, the lower third part of the face is larger than the other parts.On intra-oral examination, she had hard swelling of about 50 mm, non-pulsatile, displacement of teeth leading to malocclusion .Fig. 1PrThe panoramic dental x-ray showed a lesion of the mandible not well defined, tooth displacement, and root resorption Fig. 2TheThe computed tomography (CT) was performed and revealed an iso-dense large destructive cystic mass 34 × 43 mm in an eccentric position of the mandible, pushing the tongue back, absence of peripheral sclerosis, trabeculation, and cortical perforation .Surgical intervention was performed by the chief professor of our department.The patient has received under general anesthesia; the intra-oral approach of the cyst was done.Intra-operatively, a unilocular cystic mass without liquid content was excised.There was, however, a rupture of the external wall of the mandible cortical without iatrogenic fracture.The patient received antibiotics and antalgics daily for 8 days.The pathology of the surgical material concluded at the central giant cell tumor.Postoperative periods were favorable with the disappearance of swelling and advised to oral rehabilitation .Fig. 5PoThe patient was followed up in our specialized consultation; any clinical signs that appeared were mentioned on the patient's discharge form.Routine follow-up 3, 6, and 12 months later showed no signs of recurrence.3Common sites of giant cell tumors are the epiphysis of the long bones. Giant cell tumors accounted for 4–7% of all primary bone tumors. The mandible localization is exceptional .The most frequent clinical findings are swelling, local pain, tender mass, and pathological fractures .Aggressive lesions are defined by the presence of one or more of the following signs: pain, paresthesia, root resorption, rapid growth, cortical perforation, and a high recurrence rate after surgical curettage ,7.Differential diagnoses are aneurysmal bone cyst, chondroblastoma, giant Cell-rich osteosarcoma, and brown tumor of hyperparathyroidism ,9.In the present case patient presented with progressive swelling of the jaw with dental loss, hyperparathyroidism was excluded in this case. However, due to the exposure site, most cases of giant cell tumors disease are usually diagnosed early in childhood but in this case, the late diagnosis is caused by the lack of accessibility of the patient.The main treatment for giant cell tumors is surgical excision and different surgical approaches had been reported in the literature thus depending on the size of the cyst, localization, and experience of the surgeon .A major complication of surgical removal of the large cyst is the iatrogenic fracture of the mandible .The aggressive central giant cell tumors are less common than the literature has suggested. A strong association between multiple lesions and neurofibromatosis type 1 and Noonan syndrome has been shown to strongly associate .Given the wide range of clinical behaviors, some lesions may simply be reactive in origin while in other cases; a yet undiscovered genetic abnormality may play a contributing role.4Giant cyst tumor of the jaw is rare and less common in childhood. The main treatment is surgical removal and follow-up to reveal recurrence. Surgeons must be aware of the variety of clinical behavior of these tumors with the risk of iatrogenic fractures in large tumor surgical resection. Although they are histologically similar, it is questionable whether all GCCGs have the same etiology.The authors report no declarations of interest.The authors declared that this study has received no financial support.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.Ouassime Kerdoud: Corresponding author writing the paper.Rachid Aloua: writing the paper.Faiçal Slimani: Correction of the paper.researchregistry6312.Ouassime Kerdoud.Not commissioned, externally peer-reviewed.


Heterotopic bone formation is a rare phenomenon when associated with gastrointestinal neoplasms. Here we present a rare case of heterotopic ossification associated with goblet cell adenocarcinoma of the appendix, and a literature review of such cases associated with neoplasms within and out of the gastrointestinal tracts. We reviewed the clinical data and when available, immunohistochemical markers of osteoblastic differentiation. Our review shows similar findings to prior reports of apparent high association of heterotopic bone formation with neoplasms with mucinous features. Two, previously proposed main hypotheses of the mechanisms are reviewed. The unique feature about this case is that goblet cell adenocarcinoma was not reported previously in the setting of bone formation. Heterotopic ossification is defined as the formation of lamellar ectopic bone in the soft tissue . It is cClinical historyA 57-year-old female patient, presented with severe, intermittent abdominal pain, worsening in intensity. Abdominopelvic CT scan was performed and revealed a large pelvic mass in addition to small intestinal thickening Figure . The patSurgical intervention and gross examinationThe patient underwent an exploratory laparotomy, right hemicolectomy with en bloc omentectomy, cholecystectomy, and intraperitoneal tumor resection for her surgical management. The main specimen was received as a 30.5 cm segment of large bowel and terminal ileum with an attached appendix and a moderate amount of attached fibrofatty tissue. The appendix was adherent to the surrounding fibrofatty tissue and measured 5.0 cm in length x 1.2 cm in diameter. No tumor was grossly identified on the large bowel or ileal mucosa, or at the appendiceal orifice. Sectioning through the appendix revealed an obliterated lumen and a nodule that appeared to be contiguous with the appendiceal surface and wall. The nodule was markedly calcified and measured 2.5 cm in length x 0.9 cm in diameter and appeared to extend into the periappendiceal fat  stained slides showed a concentric transmural proliferation of tubules containing cells with minimal nuclear pleomorphism and a moderate amount of eosinophilic cytoplasm, along with goblet cells. The tumor cells were surrounded by a desmoplastic reaction and thickening of the appendiceal wall. The appendiceal lumen was obliterated with mature bone formation lined by osteoblasts. The tumor cells extended into the mesoappendix, as well as the adjacent small and large bowel, and there was an extensive perineural invasion. The goblet cells are positive for synaptophysin, chromogranin and CD56, and negative for CK7 Figure .Heterotopic bone formation is reported in association with tumors; some of which are summarized in Table  As more cases are reported, a probable mechanism for heterotopic ossification associated with neoplasia is gaining better understanding. Further studies to assess the clinical significance of bone formation in these tumors, and possible correlation with tumor behavior are warranted. One of the limitations of this case report is the lack of morphoproteomic analysis via immunohistochemistry due to financial reasons.