Schwannoma is a rare tumor that arises from the Schwann cells, which are specialized, myelin-producing cells of the peripheral nerve sheaths. As anatomic logic would dictate, these masses commonly occur in the skull base, cerebellopontine angle, and posterior spinal roots. Of this already rare entity, rarer still are the pleural schwannomas, representing approximately 1-2% of thoracic tumors. These tumors commonly affect adults with a propensity for the third and sixth decades of life and a comparative male predilection. Schwannomas are benign, indolent, and follow an asymptomatic course. As such, they often come to light incidentally.Here we report a case of primary pleural schwannomas in a 68-year-old female, found incidentally on a CT scan of the chest. To the best of our knowledge and literature review, no other similar case has been reported in our country, Pakistan. Around three weeks before her presentation, she was diagnosed with COVID-19. Her infection had run a mild course with quick recovery without the need for any hospitalization. Therefore, the manifestation of shortness of breath after resolution of all other symptoms prompted a further workup. Radiographic chest x-ray revealed an incidental finding of a large right upper lobe lung mass, slightly impinging on the trachea. This was followed by a chest CT scan at our radiological imaging facility, which showed a large, well-encapsulated, right upper lobe lung mass in the paraspinal apical location. She then underwent an image-guided biopsy of the aforementioned mass, pathological analysis of which was suggestive of a benign peripheral nerve sheath tumor (PNST) arising from the pleura . She underwent right posterolateral thoracotomy with uneventful complete surgical removal of the pleural-based lung mass. Postoperative investigations included a chest x-ray that showed interval complete resection of the mass. Currently, she is asymptomatic and her clinical condition has improved with the successful resumption of her daily routine.Physicians thus need to keep pleural schwannomas in mind as a probable diagnosis of intrathoracic tumors. Indolent and asymptomatic, they are very amenable to surgical resection with little to no chances of recurrence in the long term. However, these patients should be closely followed with repeat imaging studies when symptomatic. Schwannoma, also called neurilemmomas, are rare tumors that arise from the Schwann cells, which are specialized, myelin-producing cells of the peripheral nerve sheaths. They are usually well-circumscribed, encapsulated, and almost always solitary . FurtherAs anatomic logic would dictate, these masses commonly occur in the skull base, cerebellopontine angle, and posterior spinal roots . HoweverOf an already rare entity, rarer still are the pleural schwannomas, representing approximately 1-2% of thoracic tumors  and arisSchwannomas are benign, indolent, and follow an asymptomatic course. As such, they often come to light incidentally . AlthougHere we report a case of primary pleural schwannomas in a 68-year-old female, found incidentally on a CT scan of the chest. To the best of our knowledge and literature review, no other similar case has been reported in Pakistan.A 68-year-old woman, resident of Karachi, Pakistan, with known comorbidity of type-two diabetes mellitus, presented with complaints of mild but persistent shortness of breath on exertion, for one to two weeks. She never smoked and had no history of substance abuse.Around three weeks before her presentation, she was diagnosed with COVID-19. Her infection had run a mild course with quick recovery without the need for any hospitalization. Therefore, the manifestation of shortness of breath after resolution of all other symptoms prompted a further workup.On her clinic visit, she reported no neurological or any other systemic symptoms. On general physical examination, she was of average height and build and her vital parameters were within reference ranges. Her systemic examination revealed slightly decreased breath sounds on chest auscultation over the right upper lung field, with the remainder of the systemic examination being unremarkable. As she was symptomatic, she was admitted with the impression of post-COVID-19 sequelae. Relevant blood investigations were performed and all were within normal ranges. Radiographic chest x-ray revealed an incidental finding of a large right upper lobe lung mass, slightly impinging on the trachea , arising from the pleura, a pleural schwannoma. The patient was discussed in the multidisciplinary team (MDT) meeting and surgical resection of the mass was recommended. After a detailed discussion with the patient and informed consent, she underwent right posterolateral thoracotomy with uneventful complete surgical removal of the pleural-based lung mass. Operative findings revealed a hard, 10x10cm posterior mediastinal mass. Figure Postoperative investigations included a chest x-ray, which showed interval complete resection of the mass Figure . She is Mostly benign, PNSTs represent at least 85-90% of clinically symptomatic cases, and an even larger ratio in subclinical cases .Pleuropulmonary PNSTs tend to show the same diversity as their counterparts in more common locations and can occur as endobronchial, pleural, or parenchymal locations . Even thPleural schwannomas commonly affect adults between their third and sixth decades with males more commonly affected than females. These tumors are rather indolent and grow at an exceptionally slow rate over many years and as such rarely produce any symptoms. Discovery is often incidental on imaging studies or bronchoscopies performed for other indications. Studies have shown that up to 65-85% of pleural schwannomas are asymptomatic ,9. When Rarely, benign schwannomas may present with pleural effusions or blood-stained exudates. A recent study demonstrated an incidence of 2% for effusions in benign schwannomas . These eBenign PNSTs usually arise sporadically. However, associations with certain genetic conditions have been observed in the literature. For instance, multiple neurofibromas indicate neurofibromatosis type 1 (NF1) (Von Recklinghausen disease) and when multiple schwannomas are present, this may signify neurofibromatosis type 2 (NF2) and schwannomatosis. Malignant schwannomas have been noted to have a link to NF1; Valeyrie-Allanore et al. noted that approximately 50% of adult patients with malignant neurogenic tumors were reported to have this disease . ComparaDiagnosis of pleural schwannomas can sometimes prove to be a challenge. Advanced imaging modalities may often fail to return a definitive answer. However, they do serve to steer the conversation towards considering schwannomas. Imaging proves helpful in differentiating between isolated pleural lesions with pleural metastases, lipomas, or fibrous tumors. Kransdorf demonstrated that CT scans help to present features suggestive of schwannomas . These uConclusive diagnosis rests on histopathological analysis and subsequent immunohistochemical staining, requiring a tissue specimen . The mosThe gold standard of treatment for pleuropulmonary tumors remains surgical excision. Conservative observation has little to no justification, as primary surgical removal serves a manifold purpose, providing definitive diagnosis as well as therapy. Depending on the characteristics of the tumor, a surgical approach could involve either a minimally invasive procedure or an open thoracotomy; the precise indications of either of which are beyond the scope of this paper .Recurrence in cases of surgically resected schwannomas is a rare occurrence; accounting for less than 5% . NeitherPleural schwannomas should be kept in mind as a probable diagnosis of intrathoracic tumors. Indolent and asymptomatic, they are very amenable to surgical resection with little to no chances of recurrence in the long term. However, these patients should be closely followed with repeat imaging studies when symptomatic.


Struma ovarii is a rare dermoid tumor that consists of more than 50% thyroid tissue. The incidence of struma ovarii is reported to be 1% of all ovarian tumors and 2-5% of all ovarian teratomas. The authors present a case of struma ovarii diagnosed during the first trimester of pregnancy in a primigravida patient and discuss the clinical presentation, diagnosis, and management options for such rare tumors, both in and outside of pregnancy.  Struma ovarii is a rare ovarian tumor that can present with non-specific symptoms suggestive of a hyperthyroid state or perimenopausal symptoms in patients approaching menopause. A high level of clinical suspicion along with thorough clinical examination is crucial to diagnose such a tumor. Although benign forms are more common, malignant struma ovarii have also been documented, mainly as papillary thyroid carcinoma . Struma A 32-year-old primigravida patient of Irish-Caucasian descent visited the antenatal clinic for a booking visit on her 12th gestational week of a spontaneously achieved pregnancy. Ultrasound imaging showed the presence of a viable intrauterine pregnancy along with an incidental finding of a large 10.5 x 8.6 cm right adnexal cyst with multiple septations. The patient presented with no relevant past medical history. Her surgical history comprised of a previous laparoscopic right-sided ovarian cystectomy for a simple cyst diagnosed two years prior to her pregnancy. She had no known drug allergy and was not on any medication. She was a non-smoker with no prior alcohol use. She also had no relevant family history of malignancy.InvestigationsA repeat transvaginal ultrasound confirmed a large 10.5 x 8.6 cm right adnexal cyst with multiple septations as described previously , cancer antigen 125 (CA-125), and lactate dehydrogenase (LDH) were all within the normal range. TreatmentThe patient was started on 400 mg vaginal progesterone once daily as a vaginal pessary at the time of diagnosis at 12 weeks of gestation. Progesterone was continued until 17 weeks of gestation when the patient underwent surgery as she developed lower abdominal pain. A unilateral right salpingo-oophorectomy was performed without any complications. The left ovary was normal intraoperatively. Outcome and follow-upThe patient was followed up in the antenatal clinic two weeks after her surgery. Her postoperative course was normal, and she recovered well. She delivered her baby at 40 weeks via an emergency cesarean section due to fetal distress. The transvaginal ultrasound followed by histological examination of the patient’s presenting right adnexal cyst reported a benign dermoid cyst, confirming the diagnosis of struma ovarii Figure .Her follow-up included a yearly ultrasound scan of the remaining left ovary along with observation of her thyroid-stimulating hormone (TSH), T3, and T4 levels. The patient's plan of care for the subsequent pregnancy will be close monitoring of her thyroid function tests every trimester as well as serial ultrasound scans to detect any possible recurrence on the other ovary.The incidence of adnexal masses during pregnancy is approximately 0.2-2%, consisting mainly of benign tumors with dermoid cysts being the most common type -6. StrumThus, struma ovarii usually presents as a rare and benign ovarian mass detected as an incidental finding in pregnancy, as in this case. However, histological diagnosis is required to rule out malignant forms. In summary, a thorough history and clinical examination along with high clinical suspicion are key to recognize rare tumors, such as struma ovarii, especially in patients with symptoms of thyrotoxicosis along with a pelvic mass. While transvaginal ultrasound is the first modality of choice for investigation, diagnosis is only made on histology. The mainstay treatment for struma ovarii is suggested to be laparoscopic oophorectomy. However, ovarian cystectomy can be performed in premenopausal women with a follow-up ultrasound scan to check for recurrence.


Biliary tree neuroendocrine tumors (NET) are rare, with 100 cases in the literature, and have an excellent prognosis. Although they are rarely diagnosed before surgery, complete surgical excision offers optimal treatment.We report a case of a 36-year-old female patient referred to the surgical team with obstructive jaundice for which she was investigated and found to have a common bile duct tumor showing proximal obstruction. Excision of the tumor with hepaticojejunostomy was done. Later on, the pathology report showed grade-1, well-differentiated neuroendocrine carcinoma, which was completely excised. No further intervention was provided to the patient.Complete surgical resection with excision of the extrahepatic bile ducts and portal lymphadenectomy in addition to Roux-en-Y hepaticojejunostomy or even pancreaticoduodenectomy for distal CBD neuroendocrine tumors gives sufficient treatment in the majority of cases. No evidence of the advantage of chemo-radiotherapy as part of the treatment for this tumor.Biliary tree neuroendocrine tumors are benign tumors, and it is usually difficult to ascertain the diagnosis preoperatively. However, complete surgical excision offers an optimal treatment with no evidence of chemotherapy or radiotherapy's role in the management. •Biliary tree NETs have an excellent prognosis with a 10-year survival of 80%•complete surgical excision offers optimal treatment for biliary tree NET•Size of the tumor, presence of lymphovascular invasion, and Ki-67 assesses prognosis Commonly referred to as carcinoid tumors and usually present in the small intestine and rarely found within the bile ducts . This loPreoperative diagnosis of biliary tree carcinoid is rare; most of the cases presented in the literature were either diagnosed incidentally intra-operatively or found on the histopathology report postoperatively. Further, no evidence of the advantage of chemo-radiotherapy as part of the treatment for this tumor.Here we present a case of common bile duct NET treated with hepaticojejunostomy and found to be a NET postoperatively on the pathology report. This work has been reported in line with the PROCESS criteria .2A 36-year-old housewife, not known to have any previous chronic medical illnesses or family history of gastrointestinal malignancy, referred to our team with generalized itching that started one 1-month earlier; she also had noticed a change in urine and stool color with yellowish discoloration of her sclera. She did not document any history of abdominal pain nor weight loss. She was not alcoholic, and No previous history of surgery nor endoscopic retrograde cholangiopancreatography (ERCP) was documented. Her abdominal examination showed no palpable masses, and her liver could not be palpated with jaundice being evident.Her labs showed increased bilirubin level and evidence of obstructive jaundice, with other liver enzymes being elevated. Her CBC, INR, amylase, albumin, ESR, CRP, AMA, ANA, and tumor markers were within normal range. An initial ultrasound showed intrahepatic biliary tree dilatation with no evidence of gallbladder stones.Magnetic resonance cholangiopancreatography (MRCP) , Fig. 2 Computed tomography (CT) scan with contrast of chest and abdomen and pelvic showed enhancing soft tissue lesion in the porta hepatis with no evidence of distant metastasis. ERCP  was doneAfter the diagnosis with obstructive jaundice and the evidence of stricture in the common bile duct caused by a soft tissue lesion in the porta hepatis, biliary adenocarcinoma, and biliary stricture was the most likely differential diagnosis. The plan was discussed with the patient, and the decision was planned for surgery. Intra-operatively, a stricture along the proximal common bile duct was seen, for which the patient underwent Roux en-Y hepaticojejunostomy with excision of the tumor along with common bile duct (CBD) and gallbladder. A hepatobiliary consultant with a 20-year of experience conducted the procedure.Her postoperative course was uneventful, with improving liver function values. She was discharged home on the fifth day postoperatively. At her two weeks' follow-up, she had complete resolution of her symptoms and a standard liver function test.The pathology , Fig. 5 Later follow-up after a one-year interval did not show metastasis, and the patient was doing fine without any complications.3Neuroendocrine tumors constitute a specific histological pattern of tumors, and they were first described by Lubarsch in 1888. To this date, its origin is still unclear. It has been suggested that inflammation causes metaplasia of Kulchitsky's cells, therefore causing their malignant potential. Common bile duct [CBD] tumors represent less than 2% of all cancers, usually cholangiocarcinomas [80%]. Neuroendocrine tumors of the bile ducts account only for 0.2% to 2.0% of all gastrointestinal NET . CBD is Our patient had symptoms suggestive of obstructive jaundice, resembling most of the literature. Obstructive jaundice, biliary colic, and pruritus represent the most documented symptoms. Patients with obstructive jaundice are investigated with imaging and brush cytology, and no specific pathology could be found. This is further augmented in neuroendocrine tumors because of the submucosal location of the pathology, which yields a high false-negative result of the cytology .To this date, 150 cases of extrahepatic biliary NET have been reported. Since the first case described in 1959, only 4 cases had a preoperative diagnosis of carcinoid tumors [5.1%], with most cases being diagnosed intra-operatively or even postoperatively on the histopathology report like our case .Several reports described some characteristics to differentiate cholangiocarcinoma from NET based on history . Other rPET)/CT has no benefit in detecting these tumors [CT scan, MRCP, and ERCP are the investigation of choice for obstructive jaundice cases. NETs have a specific feature of somatostatin receptors. As a result, DOTA-TATE, DOTATOC, and DOTANOC scans can visualize the tumor. Unfortunately, this is only valid in metastatic disease . Moreovee tumors . Since oComplete surgical resection with excision of the extrahepatic bile ducts and portal lymphadenectomy in addition to Roux-en-Y hepaticojejunostomy or even pancreaticoduodenectomy for distal CBD neuroendocrine tumors gives sufficient treatment in the majority of cases ,7.The role of adjuvant chemotherapy is still questionable about NETs, as most authors could not describe any survival benefits . In the Size of the tumor, presence of lymphovascular invasion, and the quantitative assessment of Ki-67 reactive cells, Mitosis index, together with tumor differentiation, and grade help to determine prognosis, although no absolute criteria for determining the malignant potential of extrahepatic NETs have been published to this date; generally, they have a 10-year survival of 80% [Only 40% of patients with NET have hepatic metastases at diagnosis, with the liver being the most affected site . Safwan This study has multiple limitations; they include a short period of follow-up with clinic visits for one year only, and a DOTA scan was not done because the histopathology showed a low-grade, well-differentiated tumor, and it might have a role in the evaluation of metastasis.4Biliary tree neuroendocrine tumors are benign tumors, and it is usually difficult to ascertain the diagnosis preoperatively. However, complete surgical excision offers an optimal treatment with no evidence of chemotherapy or radiotherapy's role in the management.5•Neuroendocrine tumors usually present with obstructive jaundice and can be initially misdiagnosed as cholangiocarcinoma.•Biliary tree NETs are rare and have an excellent prognosis with a 10-year survival of 80%.•Although they are rarely diagnosed before surgery, complete surgical excision offers an optimal treatment with no evidence of chemotherapy or radiotherapy's role in the management.•Prognosis is assessed by the Size of the tumor, lymphovascular invasion, and Ki-67 on reactive cells.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.Not commissioned, externally peer-reviewed.No funding.This article is approved by the ethics committee of Jordan University Hospital and IRP of the University of Jordan.Mohamad Mahseer MD.: writing and editing and reviewing.Monther Altiti MD: reviewing and editing.Ahmad Jabr Al-sa'afin MD: reviewing.Tayseer Al-tawarah MD.: writing and editing.Ahmad Suleihat MD: reviewing.Saleh Abulhaj MBBS.: writing and editing.Research registration: N\A.Guarantor: Mohamad Mahseeri MD. RCSEd.No conflicts of interest.


Sinistral portal hypertension (SPH), also known as left-sided portal hypertension or segmental portal hypertension, is a rare cause of upper gastrointestinal bleeding. Historically, SPH is a result of obstruction of the splenic vein often secondary to pancreatic pathology. To our knowledge, there are no reported cases of idiopathic SPH in which the findings cannot be attributed to any etiology. It is important to do a detailed workup to rule out common pathologies of SPH before making a diagnosis of idiopathic SPH. Treatment of gastric variceal bleed secondary to idiopathic SPH can be challenging and requires a multidisciplinary approach with surgery and interventional radiology. Our patient’s history, examination findings, and imaging revealed no identifiable cause for SPH suggesting idiopathic SPH. We describe a case of isolated gastric variceal hemorrhage due to idiopathic SPH that was successfully treated. Sinistral portal hypertension (SPH) is defined as elevated pressure confined to the gastrosplenic side of the portal venous system. SPH differs from other forms of portal hypertension in that the liver function is preserved and a patent portal vein is present. It is most commonly secondary to pancreatitis, malignancy, pancreatic pseudocysts, benign pancreatic neoplasms, splenic vein thrombosis, and retroperitoneal fibrosis . SPH canThis article was previously presented at the American College of Gastroenterology Annual Scientific Meeting held in October 2020.A 53-year-old African American male with a history of developmental delay, schizophrenia, and paranoia underwent an emergent exploratory laparotomy and small bowel resection followed by a primary end-to-end anastomosis due to multiple self-inflicted stab wounds. No varices were seen in the abdomen during the exploratory laparotomy and the liver appeared normal as well. On postoperative day 10, he developed melena and epigastric pain, and his hemoglobin reduced from 10 g/dL to 7 g/dL. A repeat exploratory laparotomy and computed tomography angiogram (CTA) did not find the etiology for the bleeding. Esophagogastroduodenoscopy (EGD) was performed and showed a large isolated gastric varix (IGV-1) in the fundus and no esophageal varices. The IGV-1 had an area with active oozing. He was then emergently sent for an angiogram for possible embolization. On interventional radiology (IR) evaluation, it was noted that the portal vein and the splenic vein were widely patent, and his portal pressures were normal with a 4 mmHg hepatic venous pressure gradient, yet there were multiple large gastric varices Figure . Direct SPH is a localized form of portal hypertension sometimes referred to as left-sided portal hypertension, segmental, regional, compartmental, lineal, or splenoportal hypertension . DespiteFew case reports have demonstrated isolated gastric varices in the absence of SVT . Unlike Our patient’s bleeding was controlled with SAE which was used to block direct arterial inflow to the spleen, and thereby reduce the outflow venous pressure that results in gastric varices . AlternaSPH is a rare but life-threatening cause of UGIB that should be suspected in any patient with isolated gastric varices and normal portal pressure. Although most commonly secondary to pancreatic pathologies requiring thorough workup, it can be idiopathic. Further studies are needed to conclude if there is a congenital component to SPH that may be a contributing factor to idiopathic SPH.


Oral angiokeratoma is a rare vascular lesion that has various clinical presentations. It usually occurs as part of generalized angiokeratoma and rarely appears as a solitary lesion with no underlying systemic diseases. Only 33 cases were reported so far worldwide. In this case report, we present a rare case of isolated solitary oral angiokeratoma in a 22-year-old female patient, which is the first case to be reported in the labial mucosa that has been treated successfully by surgical excision. Angiokeratoma (AK) is a rare congenital or acquired mucocutaneous vascular lesion caused by dilated vessels lying in the superficial dermis and accompanied by epidermal reaction ,2. The eOral angiokeratoma (OAK) is a very rare lesion that usually occurs as part of a generalized angiokeratoma. However, solitary OAK that occurs as a solitary event is considered to be one of the rarest presentations of AK ,4. To thA 22-year-old female patient presented to Taibah University Dental Hospital complaining of a swelling on the left inner side of the upper lip with intermittent bleeding and painful episodes. The onset was three years ago when the patient was undergoing a fixed orthodontic treatment. The lesion started as a painful ulcer caused by repeated trauma from a sharp orthodontic appliance. A few months later, a painless papule developed. There were intermittent episodes of inflammation, pain, and bleeding upon exposure to trauma or accidental lip biting. Her medical history was free of any systemic diseases or medication consumption. Upon extraoral examination, right and left submandibular lymph nodes were palpable, mobile, and non-tender. Intraoral examination revealed a 0.8 × 0.6 cm pink, soft, non-tender, well-circumscribed papule, with some petechial hemorrhages, and it is located on the left upper labial mucosa 0.1 cm away from the vermillion border and near to the oral commissure . No otheBased on the history and clinical appearance, the preliminary differential diagnoses were irritation fibroma, neurofibroma, and pleomorphic adenoma. Since there were no signs of malignancy, surgical excision with a safety margin was performed after anesthetizing the area with 2 mL of 2% lidocaine with 1:100,000 epinephrine, to remove the lesion and establish a histopathological diagnosis. The surgical defect was closed using a 3/0 absorbable braided polyglactin suture a. The spHistological examination revealed an increase in the keratin layer, acanthosis, and papillary-like projection of epithelium. Beneath the epithelial layer, there were multiple dilated vascular vessels filled with red blood cells and surrounded by papillary projection and muscle layer . Based oIn 1889, the first case of AK was reported by Mibelli V on fingers and toes . To the As shown in , the tonClinically, OAK has various clinical presentations that can be single or multiple, reddish to bluish, and it can range in size from a couple of millimeters to several centimeters. Due to its variant clinical presentation, it can be misdiagnosed as hemangiomas, lymphangiomas, or hematomas, as well as malignant melanomas . As OAK Histologically, OAK shows hyperkeratosis and acanthosis of the epithelium with dilated vascular vessels in the sub-epithelial tissues. Cutaneous and oral AK have similar histological pictures except that hyperparakeratosis is seen in oral lesions, while hyperorthokeratosis is seen in cutaneous lesions ,11. It hOAK is believed to be due to acute or chronic trauma, high venous blood pressure, or vascular malformation. However, the exact pathogenesis is still unknown ,13. We sIn 2009, Ranjan and Mahajan proposed the first classification of OAK  37]. Ba. Ba37]. OAK is usually asymptomatic, and there is no need for any treatment once the diagnosis is confirmed, but sometimes, the lesion causes bleeding, discomfort, or cosmetic changes that necessitate intervention ,27. In aThis study has limitations. It would have been more informative to supplement histopathological examination with immunohistochemical staining for VEGF, VEGFR, MMP9, CD34, CD31, CD3, and CD8 T cell markers. Due to their unavailability, these diagnostic methods were not conducted. However, better insight could have resulted if these tests were conducted to confirm the diagnosis and exclude other causes of proliferative vascular lesions.In this paper, we reported the first case of solitary OAK of the labial mucosa that was not accompanied by any underlying systemic disorders or other cutaneous lesions. The lesion was successfully treated surgically with no recurrence detected. This case report confirms the benign nature of angiokeratoma and highlights the possibility that oral angiokeratoma can appear as an isolated mass, and the most appropriate treatment is surgical excision.


Urethral prolapse is a very rare disease, which is most often found in prepubescent girls. It occurs in about one over 5000 girls in the context of a chronic cough or any situation that increases abdominal pressure. It is often associated with diagnostic confusion, which delays management. We report the case of a 6-year-old child brought in urgently by her parents for a minimal genital hemorrhage and presence of a mass protruding from the vulva. In the hypothesis of a urethral prolapse, a medical treatment (conservative) was prescribed. After two weeks, the mass decreased significantly in volume and disappeared completely after 2 months. Urethral prolapse is the eversion of the urethral mucosa through the urethral meatus . It is aPatient information: FK a 6-year-old, student in 1st year of primary school, living in a peripheral locality of the city of Kinshasa brought by her parents for genital bleeding and the presence of a painful mass on the vulva observed a few hours before the consultation. She is the youngest of four siblings, natural born with a birth weight of 2900 grams; she followed her vaccination schedule and the Bacille Calmette-Guérin (BCG) scar is present. She presents a satisfactory psychomotor development. She was hospitalized three times for malaria and bronchopneumonia. Parents have been reporting chronic constipation for a long time.Clinical findings: the general physical examination was normal. At the genital examination, we found a reddish ovale mass of about 1 cm long axis, located just below the clitoris, sensitive to palpation, bleeding on contact. The examination under anaesthesia noted: the labia majora and minora were not oedematous, no abrasions were visualized, the hymen was present, not traumatized and the presence of a rounded mass, more or less firm to palpation, bleeding on contact, revealing the urinary meatus in the center (e center .Diagnosis: urethral prolapseTherapeutic interventions: we opted for a conservative treatment consisting of seat bath with a chloroxylenol-based solution twice a day, application of Baneocin cream twice a day, and estrogen cream twice a day. The girl also received paracetamol as needed for pain. The treatment was followed adequately as reported by the parents. There was no adverse event.Follow-up and outcome of interventions: conservative treatment was successful for our patient, but the remission was slow (was slow . After 2Parents perspectives: at the beginning, parents were suspecting a sexual abuse of their baby girl. They were reassured by the explanations received from the medical team and the evolution of the disease.Informed consent: the parents give their approval to the publication.The genital prolapse is a benign disease, the revealing mode can be different according to the cases . In our Urethral prolapse is rare and when it concerns the prepubescent girl, raises great concerns about its origin. Its rarity also induces a diagnostic difficulty for the clinician which can delay adequate management.


Plurihormonal pituitary adenomas are a unique type of pituitary adenomas that secrete two or more pituitary hormones normally associated with separate cell types that have different immunocytochemical and ultrastructural features. Although they represent 10–15% of all pituitary tumors, only a small fraction of plurihormonal pituitary adenomas clinically secrete multiple hormones. The most common hormone combinations secreted by plurihormonal pituitary adenomas are growth hormone, prolactin, and one or more glycoprotein hormones. The most common hormonal symptom is acromegaly (50%). The aim of this case report is to bring awareness about this rare type of pituitary adenomas and to describe the unique presentation of our patient, even though plurihormonal pituitary adenomas are known mostly as a clinically silent tumors.Herein, we describe an unusual case of plurihormonal pituitary adenoma with triple-positive staining for adrenocorticotropic hormone, growth hormone, and prolactin. The patient is a 65-year-old Egyptian woman who presented with mass effect symptoms of the pituitary tumor, which primarily manifested as severe headache and visual field defects. She also presented with some cushingoid features, and further analysis confirmed Cushing’s disease; slightly high prolactin and normal growth hormone levels were observed. She underwent transsphenoidal surgery and has been in remission thus far. Only a few cases have been reported in the literature, but none has exhibited silent acromegaly or mass effect symptoms as the initial presentation.This case highlights an unusual plurihormonal pituitary adenoma case with a rare combination of secreted hormones; mass effect symptoms were dominant, as were uncommon visual field defects. Our case further proves that immunohistochemical analyses of all pituitary hormones are needed to ensure correct diagnosis and to alert clinicians to the need for more rigorous follow-up due to the higher morbidity of these patients. Our case report approval number Federal Wide Assurance NIH, USA is FWA00018774 IRB registration number with OHRP/NIH is IRB00010471. Plurihormonal pituitary adenomas (PHAs) are a unique type of pituitary adenoma that are further divided into two main groups: monomorphous adenoma of one cell type producing different hormones or bi- or plurimorphous adenoma of two or more cell types producing different hormones . They reThe most common hormone combinations secreted by PHAs are growth hormone (GH), prolactin (PRL), and one or more glycoprotein hormones. The most common hormonal symptom is acromegaly 50%), followed by subclinical Cushing’s disease, then symptoms related to hyperprolactinemia [0%, folloCareful follow-up of patients with PHA is essential owing to the high risk of tumor recurrence as compared with tumors that secrete only one hormone , 6. ThisA 65-year-old Egyptian woman with a history of type 2 diabetes, hypertension, primary hypothyroidism, and class-III obesity presented acutely to the emergency room (ER) with an intractable headache and blurred vision. She had also recently experienced a worsening of her diabetes control despite being on a basal-bolus insulin regimen. She had no clear-cut cushingoid features apart from central obesity, and she had no family history of pituitary tumors or similar illness. She is a stay-at-home mother to four. Children are all a product of uneventful pregnancies and were delivered normally. She is married for 20 years to a physician. She never smoked or drank alcohol.Computed tomography (CT) brain imaging and subsequent magnetic resonance imaging (MRI) showed a sellar mass consisting of a pituitary macroadenoma that measured 2.9 × 1.5 cm invading the roof of the sphenoid sinuses and compressing the neurophysis and the optic chiasm Fig. . Intereset al. [The majority of plurihormonal pituitary adenomas produce GH, PRL, and TSH because somatotroph, lactotroph, and thyrotroph cells all arise from the same progenitor . The expet al.  recentlyet al. –23. The et al. .In the current study, we present an interesting and rare case of PHA in which the patient had ACTH-producing tumors that clinically manifested as Cushing’s disease and showed GH and PRL positivity by immunohistochemistry, both of which were asymptomatic or silent. In contrast to previously published cases where there was a predominant clinical presentation with acromegaly and symptoms of hyperprolactinemia, our patient presented initially with headaches and visual symptoms, as well as with clinical features of Cushing’s syndrome in the form of uncontrolled blood pressure, diabetes, obesity, and moon face. Postoperative pituitary MRI scans showed a successful reduction in tumor size with some residual tumor remaining in the sella, leading to the resolution of the biochemical and clinical features of cortisol excess. These rare and unusual PHAs tend to be aggressive and associated with a poor outcome . PHA patThis case highlights an unusual PHA case of a rare pituitary adenoma with cosecretion of ACTH, GH, and PRL. Mass effect symptoms were dominant, along with some cushingoid features. Although PHAs are usually clinically silent tumors, they are not uncommon pituitary adenomas, and immunohistochemical staining for all pituitary hormones is needed to ensure the correct diagnosis and to alert the clinicians to the need for more rigorous follow-up due to the higher morbidity of these patients. Further work is required to better understand the pathogenesis of this rare condition.


Fibroids of the uterus are the most common benign pelvic tumors in women worldwide. Their diagnosis is usually not missed because of the widespread and well-established use of ultrasound in gynecological clinics. Hence, the development of an unusually large myoma is a rare event, particularly in first-world countries such as Germany. It is even more uncommon that a myoma is misdiagnosed as a dietary failure.giant fibroid that reached a size of over 50 cm, growing slowly over the past 15 years, and was misdiagnosed as abdominal fat due to weight gain. We aim to discuss the factors that lead to the growth of such a huge tumoral mass, including misdiagnosis and treatment, and the psychological impact. Through this case, we intend to increase the awareness among general physicians and gynecologists. Although menstrual disorders incorporate several pathologies, adequate assessment remains the primary responsibility of health care providers. A literature review revealed approximately 60 cases of giant uterine fibroids.Herein, we report the case of a Caucasian woman with a giant fibroid can have disastrous effects on a woman’s health, including surgical trauma and psychological issues.The use of clinical and diagnostic devices, especially ultrasound, in this case, is indispensable. In conclusion, the growth of a  Leiomyomas or fibroids are the most common benign pelvic tumors in females that grow monoclonally from the smooth muscle cells of the uterus. Such tumors occur in nearly half of women over the age of 35 years, with increased prevalence during the reproductive phase due to hormone-stimulated growth . At 50 ygiant myoma is difficult with several possible differential diagnoses, the majority of uterine myomas are confidently diagnosed in the (pre-)clinical routine [giant fibroid (16.4 kg) with a size over 50 cm.In Germany, universal access to healthcare services is guaranteed by law . The Ger routine . Herein,A nulligravid, 46-year-old German woman presented to the gynecology clinic because of abnormal uterine bleeding and a slowly increasing abdominal extent in the past 15 years. She had no bowel or bladder complaints. The patient reported two episodes of polymenorrhea and menorrhagia in the past years. Due to the patient’s general fear of physicians and absence of frequent symptoms, she consulted her gynecologist and general physician sporadically. The gynecologist did not use ultrasound to clarify the uterine pathology. The general physician attributed her progressive abdominal extent to weight gain and advised dietary change and physical exercise as management. Both primary health care providers did not perform a thorough physical examination, including imaging methods, leaving the fibroid undiagnosed and untreated.2. Her abdomen was enormously enlarged and pendulous with flank fullness on both sides. An irregular mass arose from the pelvis up to the xiphisternum and was not discernible owing to abdominal wall obesity. There were no hernias or abdominal varices. Renal angle fullness was not observed. Because of the patient’s anxiety, a vaginal examination could not be performed. Transabdominal ultrasound showed a huge intraabdominal mass. The right kidney showed impaired cirrhosis, while the left kidney showed compensatory enlargement. A small amount of ascites was observed. An urgent computed tomography (CT) scan was performed revealing a large tumor that occupied the abdominopelvic cavity completely. On the CT scan, the mass measured 32 × 27 × 34 cm  and could not be visibly separated from the uterine cavity, bladder, or liver  was 32.1 kg/min situ findings, a total abdominal hysterectomy en bloc with bilateral salpingectomy was performed, and both ovaries were left intraabdominally. Postoperatively, bilateral ureteric peristalsis was confirmed. Intraoperative blood loss was 400 ml. The patient’s postoperative clinical course within 5 days of hospital stay remained complication-free with quick recovery. She was discharged after 5 days of surgery and had good overall health.A midline longitudinal incision was made from the xiphisternum to the pubic symphysis, and the abdomen was opened. A large mass arising from the uterus up to the xiphisternum, firm in consistency with enlarged superficial veins, was seen. The mass extended laterally to both flanks and occupied the right and left hypochondrium. No adhesions to the intestinal organs were observed. The bilateral ovaries were enlarged to twice the normal size, with ovarian artery pulsation seen on both sides. Additionally, the bilateral fallopian tube round ligaments were thickened Fig. a and b. Pathology confirmed a myomatous uterus measuring 52 × 37 ×  3 cm and weighing 16.4 kg. The tumor consisted of two separate myomas with diameters of more than 30 cm. Macroscopically, the shape was irregular, with overall consistency being firm with few soft areas. The tumor was pinkish-red in color, similar to (smooth) muscle cells. On the surface, enlarged aberrant blood vessels were observed. The cervix appeared normal, as well as bilateral fallopian tubes, although they were enlarged. For further histopathological examination, a cut section  was performed, and tissue sections were stained with hematoxylin and eosin and examined under a light microscope. The cut sections revealed a heterogeneous phenotype with predominant white whirling structures. Microscopically, the tumoral mass consisted of smooth muscle cells and collagen bundles. Few areas had nuclear polyploidy, blood vessels, and enlarged glands with some superficial hemorrhagic areas. There was no evidence of malignancy.giant, are exceedingly rare. The potential for benign tumors to outgrow quietly without causing specific symptoms is reasonable because of the large volume of the abdominal cavity, flexibility, and slow growth rate of the tumor [PubMed database showed approximately 60 cases of giant uterine myomas in the past 50 years worldwide [giant uterine fibroids in the past 20 years.Although uterine leiomyomas are frequent in women, fibroids > 50 cm in size, similar to the present case, with a weight of 11.6 kg 25 lb) and more being defined as 5 lb and he tumor . Online orldwide . Table 1giant myomas often undergo degenerative changes, and dystrophic calcification can complicate the diagnosis [Preoperative imaging studies are useful to define the extent of the tumor and to assess the likelihood of malignancy in cases of expansive or infiltrative growth. Ultrasonography is the preferred technique for the initial evaluation of gynecologic pathology because of its ubiquitous availability, noninvasiveness, and convenient cost–benefit ratio . In the iagnosis . Althougiagnosis . The widiagnosis , 16. Hengiant uterine fibroid a challenge. Fibroids of an enormous extent cannot be treated with the most widely used minimally invasive surgery techniques: hysteroscopic myomectomy, vaginal hysterectomy, or total laparoscopic hysterectomy (TLH)/laparoscopic-assisted supracervical hysterectomy (LASH). Similar to the present case, the majority of giant fibroids are removed during total abdominal hysterectomy with additional bilateral salpingo-oophorectomy, depending on the patient’s age and affection of both adnexa. Intraoperatively, severe complications such as hemodynamic instability can occur because of extensive blood loss [giant myomas are fatal for the patient; therefore, such patients have to be treated similarly to older multimorbid patients [Uterine leiomyomas have been misdiagnosed as adenomyosis, hematometra, uterine sarcoma, ovarian masses, and pregnancy , 17, 18.ood loss , 21. Witood loss . Generalpatients .giant fibroid development with close surveillance and early surgical therapy for women with progressive myomas is the clinical gold standard. In Germany, uterine fibroids indicate surgical hysterectomy in 60.7% of all cases [giant myoma in the present case is surprising despite the easy accessibility to professional care and high educational standard of the population in Germany. According to Stentzel et al., the utilization of professional care depends on several personal factors rather than travel time. In particular, a high socioeconomic status was positively correlated with visits to gynecological care [The prevention of ll cases . This uncal care . Data frcal care . This stGiven the patient’s unemployment for the last 3 years and her modest family background, her low socioeconomic status could have contributed to her worsening condition. Additionally, her general anxiety and previously diagnosed depressive state of mind could have led to the rejection of professional care. The misdiagnosis by her previous doctors could be explained by her lack of complaint regarding irregular menstruation. Women with fibroids of this size are expected to most likely suffer from menstrual disorders , but theGiant fibroids remain a diagnostic and surgical challenge, requiring expertise and interdisciplinary cooperation. Nevertheless, these gigantic benign tumors can be managed complication-free with proper diagnosis and surgical expertise.Preclinical utilization of the services of gynecologists in northern Germany depends on personal factors, such as family background, educational level, and socioeconomic status. Menstrual disorders are diverse in diagnosis and have organic and nonorganic reasons that require diagnostic clarification. Therefore, liberal utilization of physical and ultrasound examinations by general physicians could help prevent a delay in diagnosis and therapy of treatable causes such as fibroids.


Arachnoiditis ossificans (AO) is a rare spinal pathology that develops because of bony metaplasia secondary to chronic inflammation. AO may present with debilitating myelopathy secondary to nerve root compression, making it distinct from spinal calcification commonly seen with aging. AO is extremely rare, having been reported less than 100 times, most commonly in the thoracic spine. Even rarer still, AO has been associated with syringomyelia and arachnoid cyst because of associated cerebrospinal fluid (CSF) flow disruption. In this report, we describe a case of AO that presented with right shoulder pain, right-hand numbness, and bilateral lower extremity fatigue who had syringomyelia and arachnoid cyst discovered on MRI imaging. When brought to the operating room for syrinx shunting and cyst fenestration the dural opening was complicated by severe calcification and a diagnosis of AO was made. The patient was treated with partial resection of the calcified plaques. Syringomyelia shunting was abandoned due to low volume. Post-operatively, the patient had improvement in their myelopathy though syrinx was still visualized on follow-up imaging. This report reviews the pathology, clinical and radiographic diagnosis, and treatment strategies for arachnoiditis ossificans. Arachnoiditis ossificans (AO) is a rare spinal pathology first described in 1971 that is defined as bony metaplasia of the arachnoid membrane secondary to a chronic inflammatory state. Inflammation occurs secondary to spinal injury or manipulation though rarely it may be idiopathic. In addition, AO may be associated with the development of syringomyelia or an arachnoid cyst . TreatmeA 68-year-old female presented to the clinic with right shoulder pain, right-hand numbness, and muscle fatigue in her bilateral legs. The patient had a past medical history significant for T2-T3 diskectomy and fusion 14 years prior. Post-operative MRI three years after this procedure noted significant cord edema and arachnoid cysts causing cord compression and subsequent mild symptoms of thoracic back pain.MRI imaging that was obtained upon patient presentation demonstrated a cervicothoracic syrinx from C4-T1 as well as a thoracic syrinx spanning from T3-T10, causing significant cord expansion. Post-contrast T1-weighted images demonstrated a subtle, thin layer of contrast enhancement surrounding the thoracic syrinx. There was reportedly a diagnosis of a spinal arteriovenous malformation (AVM) at an outside hospital, and given a family history of AVMs, further vascular imaging was obtained which proved negative. The patient’s symptoms began to worsen over the subsequent months and the patient elected for operative intervention. At this time, the patient was brought to the operating room for a T2-T9 laminectomy, arachnoid cyst fenestration, and possible placement of a syringo-arachnoid shunt. Laminectomy was completed and ultrasound was used to visualize the arachnoid cyst. However, the spinal cord and syringomyelia were not able to be visualized. Dissection into the dura was difficult secondary to significant calcification. The cranial and caudal extension was performed until past the calcified portions. The arachnoid cyst was punctured with a micro-nerve hook and fenestrated. The calcified dura was carefully removed and separated from the underlying cord.Post-operative imaging demonstrated recurrent extramedullary cyst in the T3-T5 region as well as a decrease in size of the thoracic syrinx from the cervical segment to the T10 level (Figure AO is a rare spinal pathology that usually develops following spinal insult or manipulation. Inciting factors include prior surgery, infection, subarachnoid hemorrhage, lumbar epidural injections, oil-based myelography contrast agents, chemical irritation, and rarely may be idiopathic ,4. ThereAO is extremely rare, having been reported in the literature less than 100 times . In theiAO typically presents with progressive neurologic degeneration secondary to nerve root compression . The devIn cases of AO with secondary pathologies such as syrinx or cyst, ossification was identified pre-operatively in eight of 12 cases. In a case presented by Slavin et al. the diagnosis of AO was made intraoperatively, due to failure of identification on MRI and no CT on record . The stuArachnoiditis ossificans are commonly classified using the criteria laid out by Domenicucci et al., which considers morphology, clinical manifestation, and therapy . This syAO's treatment strategies have not been well-established and are typically decided on a case-by-case basis as determined by clinical presentation and morphology . MedicalIn previous cases of AO with a concomitant syrinx, the treatment is variable. It is unclear whether directly treating the syringomyelia offers any clinical benefit. Several cases with syrinx aspiration and placement of CSF shunt resulted in the collapse of the syrinx with clinical improvement ,12. OnlyArachnoiditis ossificans is an infrequent cause of myelopathy, most commonly secondary to a previous spinal insult. AO is best diagnosed using nonenhanced CT imaging though it may also be discovered intraoperatively during correction of associated pathology, including syringomyelia and arachnoid cyst. Treatment of AO is variable based on symptoms and morphology though it often includes a combination of surgical and medical management. In patients with associated syringomyelia, it is unclear whether treatment of the secondary pathology is necessary to improve outcomes.


Myxofibrosarcoma (MFS), especially radiation-Induced MFS (RIMFS) in the head and neck, is an extremely rare malignant fibroblastic tumor. The diagnosis and treatment of MFS remain great challenges. In the present study, we presented one case of RIMFS. Combined with previous literature, the clinical features, essentials of diagnosis, and treatment modalities of MFS in the head and neck were reviewed to better understand this rare entity.We reported a case of RIMFS under the left occipital scalp in a 20-year-old girl with a history of medulloblastoma surgery and radiotherapy in 2006. A total tumor resection was performed with preservation of the overlying scalp the underlying bone, and no adjuvant therapy was administered after the first operation. The postoperative pathological diagnosis was high-grade MFS. The tumor relapsed 6 months later, and then, a planned extensive resection with negative surgical margins was carried out, followed by radiotherapy. No relapse occurred in a 12-month postoperative follow-up.Planned gross total resection (GTR) with negative margins is the reasonable choice and footstone of other treatments for MFS. Ill-defined infiltrated borders and the complicated structures make it a great trouble to achieve total resection of MFS in the head and neck, so adjuvant radiotherapy and chemotherapy seem more necessary for these lesions. Myxofibrosarcoma (MFS) is a rare soft tissue sarcoma that can arise sporadically or be induced by radiation, representing approximately 5% of all sarcomas. MFS is one of the common soft tissue tumors in the extremities of elderly patients, which also occurs in the trunk 12%), retroperitoneum, or mediastinum (8%) . –30. 6–30In August 2016, a 20-year-old Chinese girl presented to our hospital with a 4-month history of finding a rapidly progressive palpable scalp swelling. Ten years ago, she was diagnosed with medulloblastoma in the fourth ventricle without leptomeningeal dissemination. Histopathologic examination revealed a classic type (WHO grade IV). Then, she received V-P shunt and surgical resection, as well as adjuvant concurrent chemoradiation .Physical examination revealed that the lesion was under the left occipital scalp beside the up end of the incision, painless, firm in consistency, and immobile. Neurological examination was unremarkable. On MR imaging, the lesion exhibited a well-demarcated hypointense mass on T1W sequences, slightly hyperintense on T2W sequences, and peripheral enhancement with obvious “tail sigh” on contrast administration . No relapse occurred in a 12-month postoperative follow-up.32MFS was first described in 1977 , the higMFS usually develops in proximal extremities of older people with a mean age of 65 years, men are usually affected slightly more often than women . MFS in MFS in the extremities usually presents as a slowly enlarging and painless mass . Due to Radiation-induced sarcomas (RIS) are increasingly seen in long-term survivors of head and neck tumors, with an estimated risk of up to 0.3%. Common histologic subtypes of RIS parallel their idiopathic counterparts and mainly include osteosarcoma, chondrosarcoma, malignant fibrous histiocytoma, and fibrosarcoma . RadiatiMRI is the most common diagnostic modality for MFS. Computed tomography (CT) is also effective, especially for those located near the air and bone. MFS has a low density on CT, a low-to-intermediate signal on T1-weighted MRI, and a high signal on T2-weighted MRI. MFS often shows abnormal signal infiltration along the facial plan on MRI that corresponds to an infiltrative growth pattern histologically, named “tail sign.” Post-contrast images can better display “tail sign” than T2-weighted images , 37. Thu39The definitive diagnosis of MFS depends on pathological examination. Histologically, a series of general parameters must be present such as spindle-shaped cells, elongated and pleomorphic nuclei, and an abundance of curvilinear vessels with thin walls and a myxoid matrix . Low-graLike other sarcomas, GTR  with negative margins remains the primary treatment for MFS . In ordeMFS is a locally aggressive tumor that has a propensity for local recurrence (LR). Even after complete resection, the risk of recurrence is still high, ranging from 16 to 57% Table . In contMFS is a locally aggressive tumor that has a propensity for local recurrence. Effective education about MFS, high-quality MRI imaging, biopsy, correct early diagnosis, and planned and wide surgical excision with negative margins are mandatory in order to provide the best results for MFS patients. Unfortunately, complex anatomical structures make MFS in the head and neck a great “challenge” to obtain a wide surgical margin. Therefore, in order to avoid local recurrence and distant metastasis, combined surgery and adjuvant chemoradiotherapy are recommended for MFS in this region. Further randomized double-blind controlled clinical trials are needed to confirm the efficacy of combined chemoradiotherapy for MSF in the head and neck.


Heterotopic mesenteric ossification is a benign bony tissue growth in the mesentery that mostly follows repetitive or severe abdominal injuries leading to reactive bone formation in the mesentery. There are only 73 cases (51 publications) identified in the literature up to the beginning of 2020.45-year-old Saudi male underwent multiple laparotomies to manage complicated appendicitis which ended with a diverting ileostomy and a colostomy as a mucus fistula. After 9 months, the patient was admitted to the General Surgery department in Al-Hada Armed Forces Hospital for an open ileostomy and colostomy reversal surgery where several irregular bone-like tissues of hard consistency and sharp edges with some spindle-shaped structures resembling needles were found in the mesentery of the small intestine and histopathology revealed of trabecular bone fragments confirming the diagnosis.The majority of cases occur mid to late adulthood with a predilection in the male gender, and usually present with bowel obstruction or an enterocutaneous fistula. Although it has no malignant potential, it may cause severe bowel obstruction that can lead to mortality, it's a rare occurrence and, therefore, is difficult to diagnose among many common abdominal disturbances.Here we report a rare case of heterotopic mesenteric ossification, which should be considered as one of the delayed complications of abdominal surgery or trauma. The time range of expecting the presentation of heterotopic mesenteric ossification following major abdominal trauma or surgery should be extended and continuously considered during differential diagnosis. •One of the delayed complications of abdominal surgery or trauma is Heterotopic mesenteric ossification.•Since 1983 there are only 73 cases of Heterotopic mesenteric ossification published to date.•Preoperative diagnosis of Heterotopic mesenteric ossification should be based mainly on the characteristic radiographic findings without relying on the levels of calcium or alkaline phosphatase.•The only way to reach the definitive diagnosis is through excision and histopathological analysis.•Heterotopic mesenteric ossification has no malignant potential, but it can cause severe bowel obstruction that can lead to mortality in already sick patients. HMOHeterotopic mesenteric ossificationHOHeterotopic ossificationCRPC-reactive proteinESRErythrocyte sedimentation rateCEACarcinoembryonic antigenCTComputerized tomographyBMPsBone morphogenic proteins1Heterotopic mesenteric ossification  is a benThe majority of cases occur mid to late adulthood with a predilection in the male gender, and usually present with bowel obstruction or an enterocutaneous fistula ,5. AlthoThis work has been reported in line with the SCARE 2020 criteria .2A 45-year-old Saudi male presented to the emergency department of a local hospital in March of 2018 with a typical picture of acute appendicitis; he was admitted for an open appendectomy. Intraoperatively, they discovered a perforated appendix; histopathology revealed a severely inflamed perforated appendix. After 4 days, his first operation was complicated by a feculent discharge from the peritoneal drain due to a complicated cecal fistula with a septic clinical picture. He was admitted for an exploratory laparotomy, and segmental resection of the involved bowel with primary anastomosis was done. Two days after the second operation, he had an anastomotic leak with peritonitis, and he had feculent discharge from the wound site and the peritoneal drain; he was shifted to the operating room for an exploratory laparotomy where a right hemicolectomy was done with primary anastomosis. On the seventh day, and despite the two operative attempts, the patient had intraperitoneal dissemination of fecal material and generalized peritonitis for the third time; he was sent for an exploratory laparotomy where a diverting ileostomy and a colostomy as a mucus fistula was done.The patient did not have any remarkable family history, he is medically free, not a smoker or alcoholic and doesn't have any significant medical history.−9/l, mostly lymphocytes (3.27 × 10−9/l). His hemoglobin was 146 g/l, platelet count was 370 × 10−9/l. C- reactive protein  normal range is 0.0 to 10.0 mm/h, and Carcinoembryonic antigen (CEA) normal range is 0.0 to 5.0 ng/ml.White blood cell count normal range is 4 to 11 × 10Pre-operative abdominal computerized tomography  with thePre-operative abdominal CT insured a patent passage of the bowel. But the calcified densities and fat stranding opacities were thought to be related to post-operative changes. Intraoperatively, laparotomy under general anesthesia showed adhesions and several irregular bone-like tissues of hard consistency and sharp edges with some spindle-shaped structures resembling needles were found on the mesentery of the small intestine . All theThe bone-like tissues were examined histologically . It show3Heterotopic mesenteric ossification (HMO) was first reported in the literature in 1983, where three patients developed heterotopic mesenteric ossification after abdominal surgery . Before HMO is extremely difficult to diagnose in patients presenting with abdominal pain and discomfort due to its rare occurrence and very low frequency worldwide. The diagnosis of mesenteric heterotopic ossification can be challenging; abdominal CT scans can help in identifying it preoperatively; however, the differentiation between dystrophic calcification, bone neoplasms, a leakage of contrast, foreign material, or extra-skeletal osteosarcoma from mesenteric heterotopic ossification can be difficult . The onlWe performed an extensive literature search of the Medline and Embase databases for articles published from 1983 up to 2020. No language restrictions were applied, and reference lists of all included studies were manually searched for other potentially eligible studies. We identified only 51 published case reports, including a total of 73 cases. One of whom was an 11-year-old child . About . The curThe time that passed from the last surgical operation to the intraoperative discovery of HMO in the current case was 9 months. The time required for the formation and appearance of HMO clinical symptoms is not exactly known but ranged from 2 weeks to 2 years . AlthougBone morphogenic proteins  are multvia primary sensory fibres to the spine and brainstem [in vitro [In our case, the patient was admitted with severe abdominal pain that reoccurred with each complication and necessitated multiple surgeries. This pain is sensed by substance P, a member of the tachykinin peptide family, that was demonstrated to transmit nociceptive sensation rainstem . This surainstem . The serin vitro .Mesenteric ossification can recure after the removal of the mesenteric bony fragments surgically; calcium and alkaline phosphatase levels can predict the recurrence. If the patient had a low calcium level and a high alkaline phosphatase level, which might indicate an ongoing process of osteogenesis and an active osteoblast . Our patAmong the 52 HMO cases presented in the literature, only five cases showed elevated levels of alkaline phosphatase, of which four cases presented 3 weeks after the predisposing trauma or surgery while the patient in the current case was admitted 9 months after the inciting operation. This might indicate the vast variation in the speed of the HMO pathogenesis from case to case, which might be attributed to the levels of inflammation during and after the surgeries, amount of released cytokine, and the ability of the body to control and adjust the orchestra of inflammation. Moreover, the pathogenesis of the HMO might be accelerated or delayed depending on the post-operative management of the case, as precise management through proper anti-inflammatory drugs might prevent or delay the pathogenesis course of the HMO. Additionally, the delayed formation of the HMO, as we encountered in the current case, might indicate the need for long-time management with continuous monitoring of the serum inflammatory cytokines even after the subside of the pain associated with the surgical operation as to continue controlling the inflammatory milieu to avoid delayed HMO formation.4In summary, here we report a rare case of HMO, which should be considered as one of the delayed complications of abdominal surgery or trauma. The time range of expecting the presentation of HMO following major abdominal trauma or surgery should be extended and continuously considered during differential diagnosis, especially when there is a history of previous surgery or trauma. Diagnosis of HMO should be based mainly on the characteristic radiographic findings without relying on the level of alkaline phosphatase, which is elevated only in the period of active osteogenic stag. Continuous monitoring and controlling of the inflammatory cytokines not only for a short time post-operatively but for an extended period may prevent or delay the HMO formation.No funding was received.The study was approved by the Research Ethics Committee at Al-Hada Armed Forces Hospital and is available upon request from the corresponding author. .Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.Not applicable.Sara Ahmad AssiriTaif University School of Medicine Taif, Saudi Arabiasaraassiriiii@gmail.comAl Qutbiyyah AT TAIF Kingdom of Saudi ArabiaNot commissioned, externally peer-reviewed.Sara Assiri and Raad Althaqafi led the writing of the case report and literature review, Rawan Aloufi, Fawaz Althobaiti, Budur Althobaiti, and Mohammad Al Adwani assisted with writing and revision of the manuscript All authors read and approved the final manuscript.No conflict of interest.


Introduction and Importance: Tuberculosis is one of the leading infectious causes of mortality worldwide. In the United States, foreign-born persons account for 70% of tuberculosis (TB) diagnoses. Comparatively, testicular cancer is much less common. However, metastatic disease may present similarly. Diagnosis is supported by elevated tumor markers and radical orchiectomy with specimen biopsy confirms the diagnosis and tumor type. Following resection, adjuvant treatment for metastatic disease includes chemotherapy.Case Presentation: This case describes a 22-year-old male immigrant with shortness of breath as the presenting symptom. Chest imaging showed a cavitary lung lesion encroaching the bronchus and left atrium. The patient was placed on airborne precautions and a complex hospital course ensued which resulted in the diagnosis of metastatic nonseminomatous germ cell tumor. The patient's 8 cm testicular tumor was treated with radical orchiectomy followed by chemotherapy. His condition deteriorated quickly, and he passed away in the hospital.Clinical Discussion: Metastatic testicular cancer is relatively rare compared to tuberculosis, especially in the immigrant population. Differentiating extrapulmonary TB from metastatic disease can pose a diagnostic challenge due to similar presentations. Complete physical exam including the genitalia is paramount in discerning a diagnosis of testicular cancer.Conclusion: Incidence of metastatic testicular cancer is much less common than extrapulmonary tuberculosis but must always be included on the differential for a young male. •Though relatively rare, testicular cancer is the most common malignancy among men 20–40 years old in the United States.•In any young man with a persistent cough, metastatic testicular cancer must be evaluated and excluded.•Complete physical exam, including genitalia, is vital following discovery of a lung lesion, especially in a young male.•Delay in diagnosis of testicular cancer may delay treatment resulting in worse patient outcomes. In thThis work has been reported in line with the SCARE 2020 criteria .2A 22-year-old Guatemalan male presented to the emergency department (ED) with left-sided chest pain and shortness of breath. He reported his symptoms began four months prior and progressively worsened. He also described intermittent hemoptysis, fevers, night sweats, and a 20 lbs weight loss over those four months. The patient had no past medical history, no history of surgeries, no known genetic abnormalities, and no family history of cancer. He had no history of drug use or allergies but had smoked cigarettes for two years before recently quitting. He had been working in construction since his arrival to the United States 18 months prior.On the initial exam, the patient was tachycardic and tachypneic. A chest X-ray revealed a right upper lobe consolidation with moderate bilateral pleural fluid. Computed tomography angiogram (CTa), conducted using LightSpeed VCT 64-slice CT system, showed large mass-like consolidation in the right perihilar region measuring 12 × 6 cm and involving the mediastinum, an area of cavitation transecting the right mainstem bronchus, and mediastinal lymphadenopathy see . An addeQuantiFERON-TB gold testing, acid-fast bacteria sputum cultures, and blood cultures were taken. Transthoracic echocardiogram (TTE) and bronchial biopsies were recommended before surgical intervention for the lung mass invading the left atrium. TTE revealed a 3.4 cm × 2.1 cm left atrial mass. Bronchoscopy showed an endobronchial mass occluding the middle and lower lobes and needle biopsies were taken. Reevaluation of the patient revealed an enlarged, nonpainful testicle. On further questioning, the patient noted the mass had been there for several months, at which point urology was consulted.Scrotal ultrasound using grey scale and color doppler revealed an 8.5 cm × 4.1 cm x 4.7 cm complex right testicular mass and a normal left testicle see . Alpha-fThe patient was started on a 5-day course of bleomycin, etoposide, and cisplatin (BEP) without further surgical intervention at that time due to the large tumor burden and cardiac involvement.The patient tolerated the treatment poorly, developing hypoxia, acute kidney failure, and neutropenia. Following completion of his 5-day BEP, a post-chemotherapy transesophageal echocardiogram and CT scan showed a significant decrease in tumor size. Despite this, the patient's condition deteriorated. Adjuvant external beam radiation was contraindicated at this time due to the patient's critical condition. He developed acute respiratory distress syndrome necessitating ECMO, then acute renal failure warranting continuous renal replacement therapy. The patient ultimately experienced acute hepatic failure and expired. An autopsy request was declined by the family. His body was transported to Guatemala for burial shortly after his passing.3This case is unique as it presents a diagnostic dilemma for the treatment team regarding an infectious versus neoplastic etiology. Given the patient's recent immigration from a TB-endemic country, infectious etiologies must be ruled out in the differential diagnosis. The patient's productive cough with intermittent hemoptysis, imaging finding of cavitary upper lobe lung lesions, and lack of enlarged retroperitoneal lymph nodes all support tuberculosis as a potential diagnosis. Reasons against possible disseminated tuberculosis include an immunocompetent state (HIV-negative) and no recent sick contacts.While TB has an incidence of 20.7 per 100,000 in Guatemala, testicular germ cell tumors are relatively rare, with rates per 100,000 of 0.6 in Guatemala and 3.9 among US Hispanics . StagingThough testicular cancer metastasis to the mediastinum is well documented, it is imperative to keep in mind extragonadal GCT without primary testicular involvement occurs in 2–5% of cases ,15. ThesThe SCARE 2020 paper was used in the development of this paper .4Symptoms of TB can cause other disease processes, such as metastatic testicular cancer, to be overlooked, posing a diagnostic challenge. Diagnosis of testicular cancer is based on a thorough history and pathologic examination. Incidence of metastatic disease is much less common than tuberculosis but must always be included in the differential for a young male. An initial more thorough examination, especially of genitalia, could have expedited the correct diagnosis and initiation of treatment, possibly saving his life.Continuing to educate young men on testicular self-examination through community outreach programs may help raise awareness for this disease.Not commissioned, externally peer-reviewed.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.


The topsy-turvy heart is a very rare cardiac malformation that involves a global 90° clockwise rotation of the heart along its long axis. This rotation results in the displacement of the great arteries and severe elongation and stretching of the brachiocephalic arteries and the bronchi. We present an unusual case of topsy-turvy heart diagnosed prenatally with a large aorto-pulmonary window and. This case gives an insight into the morphological details and clinical presentation of this rare malformation and its associated complications. We also present a review of the literature of this rare anomaly showing only 15 live cases that have been published with only three cases diagnosed prenatally. Topsy-turvy heart is a very rare cardiac malposition anomaly first described by Freedom et al. . It invoWe report a case of topsy-turvy heart prenatally diagnosed with AP window, suspected aortic coarctation, and lung deformities. We also summarize the cases reported in the literature of this rare anomaly, which include only fifteen living cases with only three cases diagnosed prenatally.A 20-year-old mother gravida 1 para 0 (G1P0) with history of congenital lobar emphysema was referred for fetal cardiac evaluation at 22 weeks of gestation because of suspected fetal cardiac anomaly with ventricular septal defect. Parents were first cousins with a family history of congenital bronchiectasis in the mother’s uncle and congenital cardiac abnormality (single ventricle physiology) in a paternal uncle. Fetal scan also revealed possible bronchial compression, single umbilical artery type 2 with persistent vitelline artery, and hypoplastic abdominal aorta. Fetal echocardiogram revealed superior–inferior ventricles with normal atrioventricular and ventriculoarterial relationships, a drop-out in the membranous ventricular septum suggestive of a defect, abnormally short ascending aorta, elongated subclavian artery, and a large AP window Figs. . PregnanThe baby was born with a birth weight of 2750 g, Apgar scores were 1, 6, and 8 at 1, 5, and 10 min, respectively. The baby was intubated right after delivery and was admitted to the neonatal intensive care unit (NICU). Post-natal echocardiogram confirmed the presence of superior–inferior ventricles. There was normal atrioventricular and ventriculo-arterial connections, a small secundum ASD, and a large AP window. There was no ventricular septal defect. Cardiac valves were morphologically and functionally normal except for a bicuspid aortic valve with no significant stenosis or regurgitation. The aortic arch was very difficult to visualize adequately due to its unusual course and tortuosity. The pulmonary artery was significantly dilated, the abdominal aorta showed significant flow reversal secondary to the AP window.Cardiac computed tomography (CT) showed upstairs–downstairs relationship of the ventricles, a distal non-restrictive AP window Fig. . The heaAngiography showed dextrocardia, horizontally superimposed ventricles with the RV superior, and the LV posterior leading to the great vessels arising horizontally, low position of the great vessels in the thorax, and a large AP window. Because of the position and orientation of the great vessels, the position, branching, and direction of the pulmonary arteries were also unusual. Similarly, branching of the aorta was uncommon with neck vessels originating low from the descending aorta with a long vertical course in the chest to the arms, neck, and head. The abdominal aorta was interrupted with highly abnormal vascularization to the abdominal organs and legs through small vessels. Post-catheterization, the baby’s clinical status deteriorated with signs of decreased perfusion requiring vasopressor support and fluid resuscitation. A flexible bronchoscopy and bronchogram showed critical stenosis with malacia of left main bronchus with pulsatile compression noted and resultant ball-valve effect on the left lower lobe Fig. .Fig. 5A A multidisciplinary team discussion led to a consensus that the airway lesion with extensive vascular airway compression and malacia were likely futile. Palliative care was offered and no intervention was performed. The baby was extubated to noninvasive ventilation and expired shortly afterward.Topsy-turvy heart, first described in 1984 by Freedom et al. , is charA review of the literature revealed a total of 17 previously reported cases with topsy-turvy heart, 3 of which were diagnosed prenatally  . Our patIn terms of outcome, 5 of the 15 live patients reported underwent surgical intervention. Four patients had AP window repair, two of which were discharged home and did fairly well based on the last follow-up reported , 9. The One patient did not require surgical intervention but required treatment for severe pulmonary hypertension . Two patThis review of this rare anomaly reveals significant complications in these patients including acute and chronic respiratory failure, recurrent lung infection, hyperactive airway and pulmonary hypertension, mostly secondary to complex airway compression. This requires immediate highly specialized medical and potential surgical care in addition to short- and long-term follow-up in these patients.In conclusion, topsy-turvy heart is a rare congenital anomaly characterized by 90° clockwise rotation of the entire heart with its great vessels around its long axis without any twisting of the atrioventricular connections. Reviewing the literature and based on our case reported above, the management and prognosis are mainly dependent on the complexity of the vascular anomaly and extension of airway disease, both of which poses major challenges in planning any surgical intervention. Prenatal detection is significantly low for this rare anomaly. In addition to its rarity, this can also be attributed to the fact that the intracardiac anatomy is usually normal as discussed while the pathology affects mainly the extracardiac structures (arteries and airways). Having said that, abnormal orientation of interventricular septum along with AP window and abnormal aortic arch on fetal echocardiogram should raise concerns for possible topsy-turvy heart. A multidisciplinary team approach, including cardiology, cardiothoracic surgery, and pulmonology is needed to guide the management of such anomalies based on each individual center’s experience.


Intracranial lipomas are extremely rare fat-containing lesions that comprise 0.1%-0.5% of all primary brain tumors. They are congenital lesions that arise due to persistence and maldifferentiation of the meninx primitive (subarachnoid space precursor). We report a case of a 30-year-old woman who presented with seizures due to an intracranial lipoma and no neurological deficits. CT (computerized tomography) imaging findings demonstrated a large interhemispheric partially calcified lipoma that communicated with a large scalp lipoma and was associated with agenesis of the corpus callosum. Compared to the prior CT imaging, the lipoma increased in size from 3.4 cm to 4.1 cm transversely. A recent CT angiogram done due to suspicion of an aneurysm showed the lipoma now measuring 6 cm by 4.7 cm. Most cases of intracranial lipoma have been reported in the pediatric age group. Here, we report a rare case of interhemispheric intracranial lipoma in the adult age group. This case also demonstrates the importance of imaging modalities for detecting intracranial lipoma without performing invasive brain biopsy. Intracranial lipomas are extremely rare fat-containing lesions that comprise 0.1%-0.5% of all primary brain tumors A 30-year-old female patient presented with a history of seizures and headaches due to an intracranial lipoma which had worsened over the past few years. The patient had a significant family history of death due to a brain aneurysm on her maternal side. The patient was initially evaluated in the emergency department for seizures 2 years ago. At that time, a CT scan without contrast was done. It showed a large interhemispheric partially calcified lipoma, which appeared to extend superiorly and anteriorly communicating with a large scalp lipoma and 3 small defects in the left frontal lobe B. FurtheLast year, the patient was seen by a neurologist for her brain mass and management of her seizures. She was unable to describe her seizures. However, she was told by witnesses that her whole body shakes during the episodes. She also explained that she has an aura before her seizures which she described as “feeling funny,” Her seizures occur once or twice a month. The patient followed up with the neurologist regarding her seizures. The patient had experienced multiple seizures over the past year. Her first seizure resulted in dizziness and her whole body shaking which caused her to bite her tongue. On the physical exam, the patient had no neurological deficits and motor strength was 5/5 in all 4 extremities. Her sensations were intact to light touch and pinprick throughout the upper extremities and lower extremities. Reflexes 2+ and symmetric throughout, no dysmetria on a finger-to-nose exam, and gait was intact. Her Levetiracetam dosage was increased to 750 mg and she was referred to a neurosurgeon.Later in the year, an electroencephalogram was performed which revealed a normal study. The patient had a follow-up with the neurologist to discuss her results. Here, the patient addressed difficulty tolerating the higher dose of Levetiracetam as it was causing headaches and panic attacks. Thus, she started to take half of the Levetiracetam and consequently continued to have breakthrough seizures. Her medication regimen was changed to Levetiracetam 500 mg in the morning and Levetiracetam 750 mg at night.The patient underwent a brain MRI with and without contrast, and a head CT scan without contrast as requested by her neurosurgeon. The head CT showed a large interhemispheric partially calcified lipoma with superior and anterior extension and communication with the large scalp hematoma in the left frontal region with an interval increase in the size of the lipoma C. WhereaSix months ago, the patient had a CT angiogram (CTA) done for suspicion of an unruptured cerebral aneurysm. The CTA revealed a large interhemispheric lipoma with agenesis of the corpus callosum communicating with the scalp. Parts of the callosal marginal branch of the anterior internal artery appeared to communicate with the diploic sinus. Most of the pericallosal artery and the internal cerebral veins runs right through the interhemispheric lipoma A. The liOxcarbazepine  was added to her current regime of Levetiracetam 500 mg. Later in the year, the patient is planning to undergo neurosurgery for resection of the large interhemispheric lipoma.Intracranial lipomas are congenital lesions because of abnormal differentiation of embryological meninx primitiva (subarachnoid space precursor). These lesions are a rare finding with an incidence of 0.08% and 0.46% in autopsy series Most intracranial lipomas are asymptomatic and found incidentally. If symptomatic, patients commonly present with seizures, but can also present with raised intracranial pressure, dementia, and hemiparesis Interhemispheric lipoma associated with a subcutaneous component is a rare finding. Most of the cases in the literature are associated in the pediatric population and only a few are found in adults –13. The Microscopically intracranial lipomas are composed typically of adipose tissue and a capsule with a variable quantity of collagen fibers, blood vessels, and calcifications Both CT and MRI findings are characteristic of intracranial lipoma, so biopsy confirmation is not required for the diagnosis. Head CT scan is diagnostic as it shows fat density attenuation (-80 to -110 HU), calcifications, location of the lipoma, and associated anomalies Intracranial lipomas in most cases are asymptomatic and do not require treatment ,6,14. Ot1.Intracranial lipomas are rare, asymptomatic, and incidental, which should be on a differential of a patient presenting with headaches or unexplained seizures.2.CT and MRI are diagnostic for intracranial lipoma, so biopsy confirmation is not required as fat density and calcification can be detected on these imaging modalities.3.Depending on size, location, vascularity involvement, and level of seizure, the patient may be potential surgical candidate.


It is extremely uncommon in the posterior mandible. It is thought to originate during the morpho-differentiation stage of dental development. However, its etiology and pathogenesis remain obscure. We report here the clinical and pathologic findings of an incidentally discovered dilated odontoma arising in the left third mandibular molar germ of an 11-year-old male and a review of the pertinent literature. As dilated odontoma is not established as an independent entity in the current WHO classification of odontogenic tumors and is the result of a well-established developmental anomaly of the tooth , it should be better identified as dilated dens invaginatus.Dilated odontoma is the most severe variant of  It may affect either the primary or the permanent teeth and its incidence has been estimated to range between 0.04% to 10%, with a greater female predilection [dens invaginatus is that proposed by Oehlers, who distinguished three different main types based on the depth of enamel-lined invagination [dens invaginatus [dens invaginatus [Dens ilection ,3. The agination . In typeaginatus ,3,6. In aginatus ,9,10,11.aginatus ,3,8. dens invaginatus, is also provided.We report here an unusual dilated odontoma incidentally detected in an 11-year-old male. The lesion was associated with the left mandibular third molar germ. A review of the pertinent literature, with a special focus on the extracted type III An 11-year-old male came to our observation for his first dental visit. His medical history was negative. No symptoms were reported by the patient or his parents. The face was symmetric and no swelling of the cervical lymph nodes was observed. Intraorally, the dentition of the permanent teeth was completed, except for the third mandibular molars and the second and third maxillary molars. Bucco-lingual expansion of the jaw bones was not evident. An orthopantomogram was performed to assess the development of third molars . Unexpecdens invaginatus cases were reported [We have presented here a rare incidentally discovered dilated odontoma arising in the left third mandibular molar germ of an 11-year-old male. To the best of our knowledge, dilated odontoma has never been reported to arise in association with the germ tooth. This makes the case reported here unique. In the most recently published review of the literature, sixteen extracted type III reported . Eight oreported ,10,11,13reported . A clinireported ,9,14. Inreported .dens invaginatus [dens invaginatus or dilated hamartoma of the teeth to identify these lesions could be more accurate than dilated odontoma.In addition to its clinical uniqueness, the case reported here offers the possibility to discuss the proper terminology to use for this type of dental lesion. In this regard, we completely agree with other authors, according to which dilated odontoma represents the extreme and most severe form of type III aginatus ,3,8. Howaginatus , dilatedaginatus ,16,17. Iaginatus ,10,11,13dens invaginatus or dilated odontoma. Although there is general agreement on its origin during the morpho-differentiation stage of dental development, as unequivocally proven in the case reported here, the fine etiology and pathogenesis remain to be elucidated. Proposed theories have included increased localized external pressure, inner enamel epithelium growth retardation or stimulation in certain areas of the tooth bud, infection, and trauma [Different theories have been suggested to explain the development of d trauma ,5,6,7,8.d trauma .dens invaginatus or dilated odontoma incidentally detected in an 11-year-old male. The lesion involved the left mandibular third molar germ. Even though the etiology and pathogenesis of this lesion remain to be defined, its origin reflects a well-defined developmental anomaly in tooth formation that is the invagination of the enamel organ into the dental papilla. As a consequence, although the existing nomenclature is more clinically acceptable, it could be simply defined as dilated dens invaginatus or dilated hamartoma of the teeth.In conclusion, the case reported here represents an uncommon example of severe type III


Spinal cord compression (SCC) is an uncommon, severe complication of Hodgkin lymphoma (HL), occurring in 0.2% of cases at the onset and in 6% during disease progression. We present a teenager with SCC with clinical onset of HL; her pre-existing neurological abnormalities covered the presence of an epidural mass, which could have misled us.A 13-year-old girl presented with a three-month history of lower back pain and degrading ability to walk. She suffered from a chronic gait disorder due to her preterm birth. A magnetic resonance imaging of the spine revealed an epidural mass causing collapse of twelfth thoracic vertebra and thus compression and displacement of the spinal cord. Histological examination with immunohistochemical analysis of the epidural mass demonstrated a classic-type Hodgkin lymphoma. Early pathology-specific treatment allowed to avoid urgent surgery, achieve survival and restore of neurological function.Children and adolescents with back pain and neurological abnormalities should be prioritized to avoid diagnostic delay resulting in potential loss of neurological function. SCC requires a prompt radiological assessment and an expert multidisciplinary management. Spinal cord compression (SCC) among pediatric patients can be attributed to various underlying conditions, including malignancy. SCC is a rare oncological emergency due to paravertebral benign or malignant tumors, arising from neurogenic, mesodermal, germ cell or lymphatic tissues , 2. Neur18F-fluordeoxyglucose positron emission tomography (FDG-PET) scan detected pathological FDG uptake involving the paravertebral tumor and the lymph nodes detected at CT scan  and C-reactive protein . A peripheral blood lymphocyte typing was negative for presence of blasts and a bone marrow aspiration allowed to exclude tumor involvement. A contrast-enhanced magnetic resonance imaging (MRI) of the brain and the spine showed an epidural mass infiltrating the spinal canal via the left neural foramen. The mass, extending from Th12 to L1 with compression and displacement of the spinal cord  , 5.SCC increases paravertebral tumor morbidity, that ranges from chronic back pain to sensory, motor and autonomic neurological disorders and paralysis , 6. FuncHL is typically nodal involving lymph nodes in 90% of cases; in 10% of cases, HL may present as an extranodal disease. SCC is a severe complication of HL reported in 6% of cases during disease progression and in only 0.2% at clinical onset , 7, 8.Cases of HL presenting with SCC have been described both in adults and children. We carried out a systematic review of the literature of all pediatric reports. We found 45 articles using the following keywords: “Hodgkin lymphoma”, “spinal cord compression”, “children” and/or “pediatric”. For each article the following restrictions were applied: publications in the last thirty years (20 articles were excluded), patient age 16 years and younger (1 article was excluded) and from publications originating in English (2 articles were excluded). Another 15 articles referred to NHL and were excluded. The remaining 7 articles consisted of: 1 systematic review, 1 review, 3 case series and 2 case reports. Those articles were analyzed to compare pediatric and adolescent patient experiences, symptomatology and histology (Table Symptoms consistent with an epidural mass depend on the level of the lesion and they may progress slowly. Prolonged back pain or discovery of radicular or nerve damage should be immediately investigated for malignant cord compression with tumor extension into the nerve roots , 9. As iMRI of the entire spine is the diagnostic radiological evaluation of choice. FDG-PET scan is essential for staging, risk stratification and identifying patients who inadequately respond to chemotherapy and should receive radiotherapy , 15.HL is a slow-growing, malignant lymphoproliferative disorder and quickly responds to initial medical therapy in most cases . CorticoMalignant SCC requires a multidisciplinary approach with experts in pediatric oncology, including a pediatrician, a radiologist, a pathologist, a radiotherapist, a surgeon, a neurosurgeon and a physiotherapist. Our expert multidisciplinary team provided initial targeted care and prevented long-term complications related to radiotherapy and surgical interventions. We carried out a minimally invasive diagnostic technique and began an early pathology-specific treatment avoiding an EDL. We achieved both survival and restoration of neurological functions.Tumors among adolescents are often missed because they are rare and adolescents, for various reasons, often do not effectively communicate their discomfort from the disease. If a tumor is suspected, adolescents should be directed to a pediatric oncology unit, where management is more age appropriate , 19.SCC is rare in patients with HL; however, it represents an oncological emergency due to consistent rates of mortality and neurological sequelae. Any physician observing children and adolescents with back or radicular pain and neurological disorders should identify SCC and suspect, beside trauma or infectious disease, an epidural mass. An urgent oncologist pediatric evaluation should be requested following SCC discovery. Prompt diagnosis is crucial and combined with the above treatment may prevent tumor mortality, SCC morbidity and avoid unintended consequences of EDL.


A gastropleural fistula (GPF) is a rare pathological connection between the stomach and pleural cavity. Diagnosis and treatment are frequently delayed due to the lack of specific clinical, laboratory and radiological findings. We describe a case of a 53-year-old gentleman who presented to our institution with respiratory sepsis and a massive haemopneumothorax on imaging. Uniquely, he was discharged a week prior after a splenectomy for a traumatic fall. Gut flora in the pleural fluid and a subsequent positive dye test suggested an aero-digestive connection. Repeat imaging revealed a fistula between stomach and the left pleural cavity through a ruptured diaphragm. He underwent an open sleeve gastrectomy and primary repair of the diaphragm. This is the first GPF in literature presenting in such a fashion. Although rare, a persistent effusion with a history of blunt thoracoabdominal trauma may herald a GPF, which, if not diagnosed promptly, may result in significant morbidity. Gastropleural fistulas (GPF) are rare pathological connections between the stomach and pleural cavity. Mechanistically, Markowttz  first deA 53-year-old gentleman was admitted after falling down a flight of stairs, landing on his left torso. Imagining identified left rib 10–12 fractures, flail chest, a left haemopneumothorax and an AAST grade-IV splenic laceration. A chest drain was inserted, and he underwent an emergency laparotomy and splenectomy with no intraoperative complications. His chest drain was removed on post-operative day three, and he was discharged on post-operative day seven after an uneventful recovery.He was readmitted three days post-discharge with worsening pleuritic chest pain and dyspnoea. On admission, he was febrile and tachycardic. On examination, he had absent breath sounds and dullness to percussion over the left mid and lower zones of his chest. Chest x-ray confirmed a large left-sided pleural effusion , which wEnterococcus faecium, Candida albicans and Escherichia coli consistent with gut flora. At this point, an aerodigestive fistula was suspected. This was confirmed by a dye test subsequently performed with green ice cream, which tainted the drainage fluid green  , 6, 7, dThis is the first case in literature reporting a GPF presenting in such a fashion. Thorough assessment and decisive action led to a satisfactory outcome. Although rare, a persistent effusion with a history of blunt thoracoabdominal trauma may herald a GPF, which, if not diagnosed promptly, may result in significant morbidity.There is no conflict of interest to report.The authors received no funding for the research, authorship and publication of this article.