Rosai–Dorfman Disease (RDD) is a rare non-neoplastic entity, also known as sinus histiocytosis with massive lymphadenopathy (SHML), characterized by a benign proliferation of histiocytes in lymph nodes. Localized forms of RDD involving the tracheobronchial tree are very rare. There is no consensus regarding the management of central airway forms and recurrence is frequent.We report the case of an 81-year-old Caucasian woman admitted in 2014 for chronic cough. Her main medical past history included a diagnosis of sinonasal RDD in 1996 with recurrent obstructive rhinosinusitis requiring repeated sinonasal surgery, and a diagnosis of tracheal RDD in 2010 with 2 asymptomatic smooth lesions (5 and 7 mm) on the anterior tracheal wall. Physical examination was normal in 2014. Pulmonary function tests showed an obstructive pattern. Computed tomographic scan revealed a mass arising from the anterior wall of the trachea that projects into the tracheal lumen. Fiberoptic bronchoscopy showed a hypervascular multilobular lesion (2 cm) arising from the anterior tracheal wall and causing 50% obstruction of the tracheal lumen. Mechanical resection with electrocoagulation of the tracheal mass was performed by rigid bronchoscopy with no complication. Histological examination demonstrated tracheal RDD. One year after endotracheal resection, the patient presented no recurrence of cough and the obstructive pattern had resolved.Reports on tracheobronchial involvement are scarce. Symptomatic tracheobronchial obstruction requires mechanical resection by rigid bronchoscopy or surgery. Recurrence is frequent, justifying long-term follow-up. Painless cervical adenopathy (87%) and impairment of general condition are the major symptoms.In 1996, a 63-year-old Caucasian woman was admitted to the hospital with sinus obstruction related to thickening and polypoid growth of the mucosa. Her medical history included ischemic heart disease and hypercholesterolemia. Treatment included lysine acetylsalicylate, celiprolol, and simvastatin. Histological examination of right and left nasal mucosa showed a histiocytic proliferation highly suggestive of RDD . From 1998 to 2003, several sinonasal surgical procedures were performed for recurrent sinonasal obstruction.In 2010, the patient (77 years) presented with isolated cough. Pulmonary function tests were normal. Computed tomography (CT) scan showed 2 nodules protruding into the tracheal lumen. Neither intrathoracic nor cervical lymphadenopathy was observed. Fiberoptic bronchoscopy showed the presence of 2 smooth lesions on the anterior tracheal wall close to the origin of the right main bronchus (7 mm) and above the carina (5 mm). Histological examination of both biopsy samples concluded on a diagnosis of RDD. Cough resolved after discontinuation of perindopril that had been recently prescribed. The patient remained asymptomatic from 2010 to 2014.1/FVC ratio of 0.40 and FEV1 91% of predicted (1.37 L). Expiratory flow-volume loop showed a reduced peak-flow and a concave appearance of the loop without reversibility after bronchodilators. Inspiratory curve showed a plateau aspect  with fever, night sweats, and weight loss.10 Axillary (23.7%), inguinal (25.7%), and mediastinal (14.5%) lymph node groups are commonly affected. Forty-three percent of patients have extranodal involvement, including skin (11.5%), paranasal sinuses (11.3%), upper aerodigestive tract (11.3%), soft tissues (8.9%), eyes (8.5%), bone (7.8%), central nervous system (4.9%), and kidneys (2.3%).12 Intrathoracic manifestations are rarely observed (2%), including pulmonary nodules or masses, interstitial lung disease, central airway involvement, and pleural effusion.15 Patients with a chronic aggressive form affecting the kidneys, liver, or lower respiratory tract may have a poorer prognosis.1RDD can occur at any age, but the disease is more common in the first or second decades of life with a mean age at onset of 20.6 years.23 and some of these cases consisted of laryngeal involvement with tracheal extension16 , or more rarely granulomatous lesions . Diagnosis can only be established on histological examination.To our knowledge, only 12 cases of tracheobronchial involvement of RDD, including our case, have been reported in the English language literature18 and second after disobstruction by rigid bronchoscopy.17 Watchful waiting was proposed in 3 cases because of incidental discovery. In 1 case, a rapidly fatal outcome did not allow any therapeutic management.15 Very few data are available concerning long-term outcome. Tracheobronchial RDD recurrence was observed in several cases. Spontaneous recovery was observed in 1 patient.13 Two deaths were reported, 1 related to RDD bronchial and pleural involvement15 and 1 due to severe pulmonary edema in a young child with neurological RDD damage.23 It is difficult to draw any definite conclusions concerning management of tracheobronchial RDD due to the small number of cases and the limited data on long-term follow-up. In our case, the patient remained asymptomatic for 4 years after the initial diagnosis of tracheal RDD, and then developed a symptomatic tracheal mass, which was successfully resected by rigid bronchoscopy with no recurrence after a follow-up of 1 year. No recommendation has been proposed for systematic bronchoscopy long-term follow-up after RDD disobstruction. Annual clinical assessment and pulmonary function tests may be useful to detect recurrence and to consider the practice of a bronchoscopy.The therapeutic management of tracheobronchial RDD required tracheostomy in 3 patients because of acute respiratory failure. Various treatments have been described, including debulking resection by rigid bronchoscopy (n = 4), laser resection (n = 2), or surgery (n = 2). Corticosteroids were used in 2 cases without efficacy, first before laser excisionIn conclusion, tracheobronchial involvement of RDD is unusual. Clinical and radiological manifestations can mimic tracheal carcinoma or other rare tumors, consequently requiring careful histological examination. The therapeutic management of tracheobronchial RDD has not been clearly defined, but may require disobstruction by rigid bronchoscopy or surgery in the presence of symptomatic tracheobronchial obstruction. Recurrence is frequent, justifying long-term follow-up.


Odontomas represent the most common type of odontogenic benign jaws tumors among patients younger than 20 years of age. These tumors are composed of enamel, dentine, cementum, and pulp tissue. According to the World Health Organization classification, two distinct types of odontomas are acknowledged: complex and compound odontoma. In complex odontomas, all dental tissues are formed, but appeared without an organized structure. In compound odontomas, all dental tissues are arranged in numerous tooth-like structures known as denticles. Compound odontomas are often associated with impacted adjacent permanent teeth and their surgical removal represents the best therapeutic option. A case of a 20-year-old male patient with a compound odontoma-associated of impacted maxillary canine is presented. A minimally invasive surgical technique is adopted to remove the least amount of bone tissue as far as possible. According to the 2005 World Health Organization classification, odontoma is an odontogenic benign tumor of the young age . DespiteCompound odontomas have numerous tooth-like structures  known as denticles. At X-ray evaluation, compound odontomas appear as well delimited lesions with a radiotransparent halo containing radiodense zones which represent small denticles, separated by fibrous septae, while in the complex types the radiodense elements appear as irregular and disorderly masses with no similarity to dental structures , 9. ThesThe aim of this case report is to describe a minimally invasive surgical procedure to remove a compound odontoma localized in the premaxilla area associated with an unerupted permanent maxillary canine. The purpose of this technique is to preserve as much as possible the surrounding bone tissue in order to promote healing and cause less discomfort to the patient during postoperative time.A 20-year-old male patient in apparently good health conditions was referred to the Odontostomatological Clinic Unit, Department of Oral and Maxillofacial Sciences , by his orthodontist for the absence of the right upper permanent canine. The subject had no significant medical history and had not reported oral trauma or infections. Intraoral examination showed the presence of the primary right canine over the physiological period of exfoliation, which meant no correspondence between chronological and dental ages Figures .A radiographic examination (panoramic X-ray) showed multiple radiopaque structures compatible with a provisional diagnosis of compound odontoma and the unerupted right canine in mesial standing .A computed tomography (CT) with the Dentascan program  was performed in order to define the extension of the lesion and the anatomical topography, showing the unerupted permanent tooth orally positioned compared to odontoma Figures .In accordance with the patient and his orthodontist, surgical removal of the odontoma and the associated impacted canine was planned. The patient received a single dose of 2 g of amoxicillin and clavulanic acid 1 h before surgery. Surgery was performed under local anesthesia . A mucoperiosteal flap was etched and raised and bone was removed on vestibular side using a low-speed dental hand-drill and a tungsten carbide bur until the crown of the permanent impacted canine was exposed . After sThe histological examination confirmed the clinical and radiographic diagnosis of compound odontoma.The postoperative period was uneventful. Postoperative treatment consisted of amoxicillin and clavulanic acid (1 g twice a day for 5 days), paracetamol , and digluconate chlorhexidine  spray.The patient was referred to the orthodontist to continue the treatment and subsequently the surgery will be programmed for implant placement.After 2 years, the implant rehabilitation was planned: the implant  was placed and traditional two-stage loading protocol was adopted. Periapical radiographs taken at the time of implant placement showed no signs of recurrence or complications at the surgical site . After tWe confirm that we have read the Helsinki Declaration and have followed the guidelines concerning this report.The term “odontoma” was introduced by Paul Broca in 1867 to describe “tumors formed by the overgrowth of transitory or complete dental tissues.” Odontomas are intraosseous lesions mainly located in the anterior maxilla and anterior mandible, although lesions localized in gingival soft tissues have also been reported . The majThe treatment of choice for compound odontomas is surgical removal, followed by histopathological analysis to confirm the diagnosis , 22–25. In this case, the removal of odontoma was followed from the extraction of the deciduous and permanent canines. The surgical difficulty was determined by necessity to adopt a technique as much conservative as possible in prediction of the subsequent intervention of implant insertion. Therefore, the bone tissue removal around the wound was minimized, making the surgical steps of lesion and tooth removal more complicated.A careful evaluation with panoramic and CT Dentascan X-rays revealed a buccal position of the permanent canine to the odontoma. For this reason, a double access was chosen, creating two small bone gaps with the use of low-speed dental hand-drill and a tungsten carbide bur to extract the components from two different small sites instead of a single large one. This conservative approach allowed saving precious bone ridge and avoiding the formation of tissue defects; therefore it was not necessary to use filler materials or to perform guided bone regeneration procedures unlike other cases reported in the literature .A histological analysis was finally taken in order to confirm the odontoma's diagnosis.In conclusion, the presence of odontoma in association with the impacted canine needs an early diagnosis and a surgical removal treatment. A careful knowledge and an excellent evaluation of X-ray documents are essentials to resolve adequately each clinical case. The adoption of a conservative surgical approach is advisable, in order to preserve the dental tissues and obtain optimal tissue healing. A histological evaluation is necessary to confirm the correct diagnosis of odontoma.


Intradural tumours of the spine are usually benign and have a good prognosis, if they are diagnosed and removed early. Lumbar disc herniation is a common cause of chronic, acute, or recurrent lumbar radiculopathy. However, to date, there have been no reports of progressive neurological deficiencies due to the co-existence of two significant pathologies contributing to intradural and extradural compression. The current study reports the rare case of a patient with simultaneous extradural and intradural compression of the nerve root due to co-existent intervertebral disc herniation and an intradural schwannoma at the same level. A 71-year-old female suffering from lower back pain and radiating pain of the right lower extremities was admitted to Busan Korea Hospital . Magnetic resonance imaging revealed lumbar disc herniation at L4–5 and a mass occupying the intradural space at the same level of the compressed dural sac. Using the posterior approach, surgical excision of the two pathologies was performed. Pathological diagnosis confirmed schwannoma and the symptoms markedly improved. Lumbar disc herniation is the most common cause of lumbar radiculopathy . The majA 75-year-old female had been experiencing lower back and right lower extremity pain since May 2011, which led them to seek help from local medical clinics. The patient’s symptoms were occasionally alleviated by medication and physiotherapy.In January 2013, the patient visited an orthopedist. Plain films of the lumbar spine revealed lumbar spondylosis, which did not improve with pharmacotherapy. The patient then underwent a magnetic resonance imaging (MRI) scan of the lumbar region, confirming the diagnosis of spinal stenosis and intradural tumor. An incidental intradural structure, which following gadolinium injection became increasingly distinct, was noted and diagnosed as a schwannoma . The patIn May 2013, the patient’s lower back pain worsened suddenly and the patient experienced numbness and motor weakness of the right lower extremities. Neurological examination revealed weakness of the extensor hallucis longus and the gluteus medius. The patient reported abnormal sensations in the right buttock and lateral leg, however, bladder and bowel function were normal, and no reduction in deep tendon reflex was identified. The results of the straight leg-raising test were 40°/70° and the Patrick’s test results were normal. These observations led to a suspected diagnosis of a rapidly growing tumor with right L5 radiculopathy.However, follow-up MRI scan revealed lumbar disc herniation compressing the thecal sac and right neural foramen at L4/5, and an intradural structure at the same level . To remoThe pathological report confirmed the tumor to be a schwannoma . The neuThe present case report describes a patient with intradural schwannoma whose symptoms initially improved with conservative treatment, but later worsened with neurological deficit. Further investigation using contrast-enhanced MRI incidentally revealed disc herniation at the site of the intradural tumor, which was exacerbating the pre-existing symptoms.Coexistence of a lumbar disc herniation and a proximal dorsal or cervical intradural tumor has not been reported in cancer patients . The simThe majority of intradural tumors follow a slow progressive, indolent course and, thus, there may be lag period of several months prior to the symptoms becoming evident ,9. LowerThe detection of small and slow-growing schwannoma accompanied by lumbar HIVD requires the use of MRI with contrast enhancement. Although schwannoma and herniated IVD appeared bright on T2-weighted MRI, only the schwannoma was shown by gadolinium and, thus, the use of different contrast enhancement may be used to distinguish between the two .The preoperative detection of a schwannoma exhibits a significantly positive impact on the outcome of surgery and postoperative recovery. Spinal nerve compression due to tumors or HIVD cannot be easily distinguished clinically. Therefore, a complete history must be obtained and physical examination in conjunction with investigative measures, such as MRI with contrast, must be performed to ensure early diagnosis and implementation of adequate treatment. Furthermore, physicians must not confirm the diagnosis without performing full adequate studies.


Bladder schwannomas are exceedingly rare, benign or malignant, nerve sheath tumors that are most often discovered in patients with a known diagnosis of Neurofibromatosis type 1 (NF1). A few sporadic case reports of bladder schwannoma have been published in urologic, obstetric/gynecologic, and pathologic journals. We report a case of an isolated schwannoma of the urinary bladder. To our knowledge, this represents only the sixth case of benign schwannoma of the urinary bladder in a patient without von Recklinghausen disease. Schwannoma of the urinary bladder is an extremely rare tumor. It arises from Schwann cells in nerve sheaths and may be malignant  or benigWe report the case of a female patient aged 50, with no particular history admitted to our department for management of hematuria.Her history of the disease was within 3 months of the onset of intermittent total hematuria associated with irritative lower urinary tract signs namely urgency and voiding burns. Clinical examination was normal, apart from hypogastric tenderness.Before the clinical abdominopelvic ultrasound was asked a table, she showed a Right and left kidney of normal size, and bladder smooth contour seat was a mass of tissue echo structure (58 * 52 * 15mm) of the postero -lateral right . LaboratThe cystoscopic exploration objectified non- papillary solid tumor of the left bladder trigone and lateral side, both ureteral meatus were free. Biopsies of the lesion were sent to pathology for further evaluation .On macroscopic examination, the submucosal mass appeared tan, smooth and rubbery. The mass was sectioned and stained with Hematoxylin and Eosin (H&E) for further evaluation. Light microscopy revealed a spindle cell neoplasm with areas of hypocellularity (Antoni B) and areas of dense cellularity (Antoni A). Within the densely cellular areas, palisading nuclei alternated with pink, nuclear free zones (Verocay bodies). These findings are highly characteristic of a schwannoma.. Immunohistochemical study showed that tumor cells express the anti PS100 Ac, which confirms the final diagnosis of schwannoma of bladder  .CT of the abdomen and pelvis was performed documenting no evidence of tumor extension outside the confines of the bladder; the mass was well circumscribed with a density of 32 Hounsfield units (HU) on the left side of the bladder wall. The mass was isodense relatively to the wall of the bladder and measuring 5.4 X 6.1 cm of axial dimensions .The final treatment consisted of the production of a transurethral resection of bladder with good control of the tumor. The patient had no immediate post-operative complications. She was followed for 5 months without tumor recurrence before moving out of state.It was in 1910 that VEROCAY described the first case of nerve sheath tumor and gives it the name of neuroma. In 1932, Masson .Schwannoma is a ubiquitous lesion may sit in any part of the body where a nerve sheath is present, including the bladder. ROBERT  giving tClinical symptoms are nonspecific, most often provide hematuria and irritative lower urinary tract seats. Nevertheless, additional examinations of imaging are needed to advance in the diagnosis .Ultrasound has give a notion of mass is now widely supplanted by scanner (CT) and Magnetic resonance imaging (MRI). Both tests give a clear idea of the location of the tumor . The CT The diagnosis can only be made by histology. Two highly characteristic patterns for schwannoma can be seen on H&E staining. Antoni A areas consist of compact intersecting spindle cells with elongated nuclei arranged in parallel bundles, incomplete whorls and complete whorls (Verocay bodies) . Antoni Treatment of these tumors has included cystectomy, transurethral resection, observation, radiotherapy, chemotherapy, and urinary diversion. The current belief regarding treatment is that in the absence of other sequelae, these neural sheath tumors can be treated conservatively .The prognosis is related to the risk of recurrence, which is extremely rare, if the exeresis is complete. The Benin schwannoma rarely escalates. One case has been described  or maligBladder schwannoma is a very rare tumor whose diagnosis is histological. Treatment of this tumor remains uncodified, but a full excision of the tumor is mandatory to avoid recurrence. Although the degeneration of the tumor is exceptional surveillance by an annual scanner is legitimate.


Aim. Intravenous leiomyomatosis (IVL) with cardiac extension (CE) is a rare variant of benign uterine leiomyoma. Incomplete resection has a recurrence rate of over 30%. Different hormonal treatments have been described following incomplete resection; however no standard therapy currently exists. We review the literature for medical treatments options following incomplete resection of IVL with CE. Methods. Electronic databases were searched for all studies reporting IVL with CE. These studies were then searched for reports of patients with inoperable or incomplete resection and any further medical treatments. Our database was searched for patients with medical therapy following incomplete resection of IVL with CE and their results were included. Results. All studies were either case reports or case series. Five literature reviews confirm that surgery is the only treatment to achieve cure. The uses of progesterone, estrogen modulation, gonadotropin-releasing hormone antagonism, and aromatase inhibition have been described following incomplete resection. Currently no studies have reviewed the outcomes of these treatments. Conclusions. Complete surgical resection is the only means of cure for IVL with CE, while multiple hormonal therapies have been used with varying results following incomplete resection. Aromatase inhibitors are the only reported treatment to prevent tumor progression or recurrence in patients with incompletely resected IVL with CE.  Intravenous leiomyomatosis (IVL) with cardiac extension (CE) is a rare variant of otherwise usually benign uterine leiomyoma. Complete surgical excision is the only means of curative treatment. However this may not be technically feasible or individually desirable from the patient's perspective with combined thoracoabdominal operation usually required to achieve complete resection. Given the high recurrence rate following incomplete surgical resection, the use of adjuvant hormonal therapy has been investigated in an attempt to control or eradicate remnant disease. These agents have been used with varying success in patients with incompletely resected disease. This review discusses the use of hormonal treatment of IVL with CE, with two cases demonstrating the effective use of aromatase inhibitor therapy in both the short and long term when complete surgical resection is not achieved.Electronic databases were searched for the terms “intravenous leiomyoma” OR “intravenous leiomyomatosis” OR “intravascular leiomyoma” OR “intravascular leiomyomatosis” AND “cardiac extension” OR “extension to the heart” OR “intracardiac” OR “intracardiac extension”. These results were then individually searched for cases of hormonal or medical treatment following incomplete resection. The term “benign metastasizing leiomyoma” (BML) was also included in our literature search as this pathology is similar to IVL and some authors describe treatments for both BML and IVL in their series. Each article was then individually reviewed to confirm cases or reports of cardiac extension of IVL. The references of the articles retrieved were also reviewed in order to identify any further relevant studies. All studies including case reports, case series, and original articles were included. Studies were limited to those published in the English language.The cardiothoracic database at our institution was searched for any patients who had undergone sternotomy for resection of intravenous tumors with cardiac extension, from the inception of our unit database to the present. The cases identified had their pathology reports checked to identify the presence of IVL with CE. The medical records of these patients were accessed to identify the degree of surgical resection and any treatments given following incomplete resection. Outcomes were determined from specialist follow-up reports.All articles identified were case reports or case series. All available reports of hormonal therapy for IVL with CE are included in this review. Two articles reported on both BML and included cases of IVL in their reports. Five articles included literature reviews of IVL with CE. These studies discuss diagnosis, treatment, in particular surgical strategies, and outcomes. Hormonal therapies have included progesterone, estrogen modulators, hypothalamic-pituitary axis suppression of gonadotropin-releasing hormone, and estrogen antagonism. While the use of these hormonal agents as therapy following incomplete surgical resection is discussed in individual studies, there is currently no study investigating the outcomes for each individual hormonal agent.Two cases were identified from our records of incomplete resection of IVL with CE. Both patients received hormonal therapy following surgical treatment. They are included in this review as their results contribute significantly to the available literature.A 53-year-old female was reported to have a free-floating thrombus in her inferior vena cava (IVC) identified on computed tomography (CT) of the abdomen, eight weeks following a right oophorectomy for an ovarian mass. The thrombus extended from the right external iliac vein to the level of the hepatic veins . She hadA 62-year-old female was found to have bilateral pulmonary emboli on computed tomography pulmonary angiography (CTPA) following two episodes of syncope . She undUterine leiomyoma is the most common benign pelvic tumor in women , 2. DireIntravascular extension with cardiac involvement was first reported in 1907 , 12, 13.The first successful operative resection of IVL with CE was reported in 1980 . Since tSecondly, the intracardiac portion of the tumor should be urgently resected if the patient presents with any signs of cardiac obstruction or failure , 7. HoweFinally, either a single- or multistage operative approach can be used with similar low intra- and postoperative complications. The urgency, surgical approach, and combination or separation of abdominal and thoracic procedures depend on the presentation, extent of the tumor, patient factors, and surgical services available , 23. A sThe use of hormonal therapy in IVL with CE was initially trialed on the theoretical basis that the tumor has similar hormonal characteristics to myometrial tissue and uterine leiomyoma . If effeHormonal therapies used in IVL with CE aim to induce a state of systemic hypoestrogenism, thereby starving tumor cells of their hormonal growth stimulus. Early reports of postoperative hormone therapy after complete surgical resection recommended against their use as side effects are common due to the systemic nature of hormonal treatment as well as the realization that complete surgical excision offered curative treatment. This meant that these treatments were no longer needed following surgery in most reported cases. Recent reviews of IVL with CE treatment state that the use of hormone treatments such as Tamoxifen, Letrozole, or Methoxyprogesterone does not offer survival benefits or prevent tumor recurrence following incomplete excision , 7. Not The first hormonal therapies for uterine leiomyoma were reported in the early 1980s. Leiomyoma expresses both estrogen and progesterone receptors, with estradiol able to induce progesterone receptor expression and support progesterone action on leiomyoma tissue . In uterSERMs are nonsteroidal estrogen receptor ligands that display tissue specific agonist-antagonist estrogenic action . LeiomyoAn analysis of 194 cases of IVL with CE concluded that postoperative antiestrogen therapy using Tamoxifen did not prevent tumor recurrence . StudiesIn addition to these treatment results, SERMs such as Tamoxifen are also not recommended for use in women with a previous history of thrombotic events such as deep venous thrombosis (DVT), stroke, and pulmonary embolus (PE). Considering that many patients with IVL with CE present with symptoms suggesting PE or DVT, this treatment is not appropriate for these patients. The use of Tamoxifen in our first case essentially demonstrates a similar response to many other documented cases of its use in IVL with CE. It may have restricted the progression of the tumor initially; however this may also simply be attributable to the slow progressive nature of the tumor. After one year of treatment with Tamoxifen, new pulmonary lesions had developed and treatment was switched to an aromatase inhibitor. Tamoxifen also has a range of adverse effects, which contribute to the unsuitability of long-term antiestrogen therapy and were experienced by our first patient. Primarily, agonistic effects on the estrogen receptors of the endometrium increase the risk of endometrial carcinoma , as wellThe use of GnRH agonists has been increasingly investigated for the treatment of uterine leiomyoma, as well as breast and prostate cancer. GnRH agonists are a synthetic peptide modeled after the hypothalamic neurohormone GnRH which interacts with the receptor to release pituitary hormones FSH and LH, causing systemic estrogen and progesterone suppression . ThroughThe main concerning features of long-term therapy with GnRH agonists are a reduction in bone mineral density caused by low circulating estrogen levels , 36. OthAromatase catalyzes the final step in estrogen synthesis, converting the androgens androstenedione and testosterone to the estrogens estrone and estradiol . AIs areOnly a single case report for AI use in IVL with CE is available in the current literature. This report by Biri and colleagues describe the use of Letrozole in a 39-year-old woman who had pelvic recurrence of incompletely resected IVL . After 6Our two cases represent the only other reports of AI use in IVL with CE and the first reports of the use of Anastrozole specifically. In our two cases, the tumors of both patients had strong estrogen receptor positivity confirmed on histological staining. While long term follow-up is not yet available for our second case, Anastrozole use has proven to be an effective therapy in our first case, with no tumor growth of either lung nodules or intravenous tumor recurrence over a ten-year period.Bone mineral density loss and vasomotor symptoms such as flushes are the most commonly reported side effects of AIs. However, these are reported less frequently than with GnRH use . The mecSurgical excision of IVL with CE offers curative treatment and can be performed using either a single- or multistaged operative approach. If complete resection is not achieved ongoing treatment with hormonal agents should be considered as the recurrence rate can be over 30%. The use of hormonal therapies traditionally used in uterine leiomyoma for suppression of the estrogen-sensitive growth of IVL with CE has yielded mixed results. Aromatase inhibitors are a relatively recent treatment addition to the hormonal blockade of estrogen-dependent tumors. These agents can potentially provide patients with incompletely resected IVL with CE, with progression-free survival in the short to medium term.


A twenty-five-year-old female presented with a large retroperitoneal mass. Workup included history and physical exam, imaging, biopsy, colonoscopy, and gynecologic exam. After surgical resection, the mass was determined to be a primary retroperitoneal mucinous tumor (PRMT). Clinically and histologically, these tumors are similar pancreatic and ovarian mucinous neoplasms. PRMTs are rare and few case reports have been published. PRMTs are divided into mucinous cystadenomas, mucinous borderline tumors of low malignant potential, and mucinous carcinoma. These tumors have malignant potential so resection is indicated and in some cases adjuvant chemotherapy and/or surveillance imaging. This is the case of a twenty-five-year-old female who presented with a very large retroperitoneal mass, which was eventually diagnosed as a primary retroperitoneal mucinous tumor. Because this is a very rare tumor, few case reports exist describing this pathology. These tumors can be easily confused with more common tumors of pancreatic and ovarian origin. This case report provides a reminder that primary retroperitoneal mucinous tumors should be included in the differential diagnosis of retroperitoneal masses.A twenty-five-year-old female presented to her student health clinic with increasing abdominal fullness, left-sided lower back pain, and left lateral thigh numbness that had been worsening over the course of several months. Family and medical history were unremarkable. Physical exam was significant for a large fixed mass in the left upper and lower quadrants of the abdomen. She had an elevated carcinoembryonic antigen of 49. Computed tomographic scan (CT) showed a large retroperitoneal mass with cystic and solid components .The patient underwent ultrasound-guided biopsy of the solid portion of the mass. The specimen  was consistent with well-differentiated adenocarcinoma, with intestinal morphology.The patient subsequently underwent a colonoscopy and a gynecologic exam, neither of which revealed any abnormalities. Positron emission tomography scan showed avidity of the primary lesion but no distant metastatic disease.A referral to our center was made. The patient underwent a laparotomy and resection of a well-encapsulated 21 × 15 × 13 cm retroperitoneal mass . The tumThe mass consisted of a cystic cavity lined by complex tubulopapillary formations composed of columnar cells with high-grade nuclei, multinucleated cells, and frequent atypical mitotic figures. Pathology determined that the mass was most consistent with a primary retroperitoneal mucinous tumor (PRMT) with an associated invasive moderate-to-poorly differentiated adenocarcinoma .Primary retroperitoneal mucinous tumors are extremely rare with only a few cases reported in the literature. These tumors usually present in premenopausal women, though it has also been described in postmenopausal females and men . The chiHistologically, PRMTs are divided into mucinous cystadenomas (MC), mucinous borderline tumors of low malignant potential (MLMP), and mucinous carcinomas (MCa). In a recent case series, half of the MLMP and MCa cases revealed goblet cells, consistent with intestinal differentiation and ovarian stroma . This hiThere are a number of  PRMT case reports in the literature and few report outcomes. Prognosis seems to correlate with histologic grading of the disease with MC tumors having a better prognosis than MCa tumors. One case series provided follow-up results for sixteen of eighteen cases. Follow-up ranged from 1 to 148 months. Of the sixteen, two patients died from the disease, and both had MCa with anaplastic carcinoma or sarcoma. The fourteen remaining patients were alive and only three of them had known disease at the time of follow-up [It should be noted that consideration was given to the diagnosis of a pancreatic type mucinous cystic neoplasm (MCN) in our patient's case. It was postulated that the tumor may have originated from ectopic pancreas tissue or arose in the tail of the pancreas and then separated. However, there was no normal pancreatic tissue in the specimen nor were there any obvious abnormalities of the pancreas during surgery.Pancreatic mucinous cystic neoplasms (MCN) are defined by the WHO as “cystic epithelial neoplasms that occur almost exclusively in women; do not communicate with the pancreatic ductal system; and are composed of columnar, mucin-producing epithelium, supported by ovarian-type stroma .” As allChemotherapy and radiation therapy can be considered in those patients whose tumors display malignant components, as was the case with our patient. Follow-up should consist of surveillance imaging unless the tumor is found to be benign, as PRMTs have been reported to recur both locally and distally. Our patient received four months of cisplatin and gemcitabine. One and a half months after initiation of chemotherapy, our patient was found to have an iliac bone metastasis. This was treated with stereotactic radiation therapy in addition to the adjuvant chemotherapy. She currently has no evidence of disease and is undergoing surveillance imaging.


Benign pneumoperitoneum in newborns is not a rare condition that should be managed conservatively. Neonatologists and surgical teams should work together to avoid unnecessary and potentially risky procedures. Neonatal pneumoperitoneum is a well‐known emergency that is usually related to necrotizing enterocolitis in preterm infants Idiopathic pneumoperitoneum in neonates is not a rare condition reported in one series in 6% of cases with pneumoperitoneum In this study, we present a case of a newborn who developed pneumoperitoneum. The patient was managed by exploratory laparotomy and was found to have no gastrointestinal perforation, but developed severe complications post surgery. We propose a criterion to differentiate between surgical and benign neonatal pneumoperitoneum.A 1‐day‐old male newborn (34 weeks of gestation) delivered by cesarean section with a birth weight of 2280 g had APGAR scores of 8 and 9 at 1 and 5 min, respectively. The infant was admitted to the NICU with respiratory distress syndrome and was managed by noninvasive positive pressure ventilation (NIPPV). The chest X‐ray showed severe perihilar infiltrates bilaterally with a fine granular pattern [Fig. Urgent laparotomy was done. There was no gastrointestinal perforation, and the bowels were grossly healthy. A few hours post surgery, the infant developed metabolic acidosis, followed by hypotension and delayed capillary refill. He was managed with inotropes and intravenous hydrocortisone. A head ultrasound was done, which showed bilateral grade 3 intraventricular hemorrhage. The infant had a severe drop in hemoglobin level that necessitated blood transfusion. This was later complicated by posthemorrhagic hydrocephalus that needed VP shunt insertion. The infant also developed a large ductus arteriosus, which responded well to fluid restriction and a 3‐day course of oral paracetamol.A follow‐up abdominal X‐ray showed no free air in the abdomen. The results of the abdominal examination remained normal, and the infant passed meconium on the second postoperative day. After he was taken off the inotropes, gradual enteral feeding was started, which the infant tolerated very well.Neonatal pneumoperitoneum with no identified cause is not a rare condition. Increasing the awareness of neonatologists and surgeons about this condition can save newborns from surgical complications and severe neonatal morbidities.The infant in this study had major postoperative complications, including grade 3 intraventricular hemorrhage and a large ductus arteriosus. Both complications are not common for this gestational age and weight. Although the respiratory condition of the infant might have potentiated his risk for such complications, general anesthesia, hypothermia during surgery, intraoperative fluid boluses, and the transfer process between the NICU and the operating room could have added considerably to the risk of such complications.Most pneumoperitoneum cases result from gastrointestinal perforation; thus, exploratory laparotomy is most often indicated. However, differentiating benign from surgical pneumoperitoneum might be the key to saving a particular infant from severe morbidities.We propose, first, that all the nomenclature of the condition be changed from idiopathic or spontaneous pneumoperitoneum to benign pneumoperitoneum in newborns to remind physicians of the management and clinical course pattern of this condition. We further suggest the inclusion of cases in which a thoracic air leak is evident and those that are considered idiopathic because both cases are benign. In addition, we propose a criterion to differentiate those from cases due GI perforation Table .Neonatologists and surgeons should first focus on the clinical status of the newborns and the results of the abdominal examination. Then, they should try to distinguish between those newborns with risk factors for gastrointestinal perforation and those without. However, newborns without any apparent risk factors can still develop severe gastrointestinal complications. The authors reviewed the literature for cases with idiopathic pneumoperitoneum and for those due to intestinal perforation. Common characteristics for each condition were identified. The proposed criterion is only a guide and a reminder of the process that should be applied to make the right treatment decision for newborn infants.A gestational age of <32 weeks, a birth weight of <1500 g and enteral feeding are well‐known to be risk factors for necrotizing enterocolitis Benign pneumoperitoneum in newborns is not a rare condition that should be managed conservatively. Neonatologists and surgical teams should work together to avoid unnecessary and potentially risky procedures. Because there are no diagnostic tests for this condition, the use of the proposed criteria should guide the treating physician when dealing with neonatal pneumoperitoneum.None declared.This is a case report. No patient identification was used. No ethical approval was needed.


Hamartomas are very rarely identified as an endobronchial lesion.Herein, we describe a peculiar case of a 55-year-old woman with persistent cough and increasing dyspnea and radiological detection of a solid lesion subtotally occluding the main right bronchus. Despite the radiological and radiometabolic (18-fluoro-2-deoxy-d-glucose positron emission tomography/computer tomography scan) features were highly suspected for bronchial carcinoid, the definitive diagnosis after endoscopic removal was indicative of an endobronchial hamartoma.When considering differential diagnosis of an endobronchial lesion, the physicians should take firmly in mind such rare entity and, accordingly, bronchoscopy and bronchoscopic biopsy should be done as first step in management of all cases presenting with endobronchial lesions. Differential diagnosis of endobronchial tumors encompasses benign  and malignant lesions , metastasis, mucoepidermoid, or adenocystic carcinoma), generally considering first the bronchogenic carcinoma in the differential diagnosis of malignant endobronchial lesions.1 Herein, we describe a peculiar case of an endobronchial hamartoma (EH) subtotally occluding the right main bronchus and mimicking BC tumor.Hamartomas are rarely identified as an endobronchial lesion; in the largest review series that has been published (215 patients), only 1.4% of hamartomas had an endobronchial location, and most of them were not associated with respiratory symptoms.d-glucose positron emission tomography/computed tomography (18F-FDG PET/CT) scan was planned for staging and diagnostic purposes. A mild uptake of the tracer was detected at the level of the endobronchial lesion  = 2.1, Figure A 55-year-old Caucasian woman with an unremarkable past medical history presented at Emergency Department for persistent cough and increasing dyspnea. Physical examination revealed decreased breathing sounds in the lower part of the right hemithorax. Chest x-ray Figure A revealeAfter multidisciplinary consultation, despite considering any risky procedure, an endoscopic removal was planned as first step approach with the double aim of solving the bronchial obstruction and obtaining a definitive pathological diagnosis. Despite we taken into account a 1,4,7,10-tetraazacyclododecane-N,N′,N″,N″’-tetraacetic acid (DOTA)-peptide PET scan evaluation for better characterizing the endobronchial lesion, we decided to perform directly the endoscopic removal of the lesion because the increasing dyspnea was becoming clinically significant (few violent episode of cough) and in consideration of the time required for scheduling such radiometabolic evaluation.Rigid bronchoscopy showed complete obstruction of the mid-portion of the right main bronchus caused by a polypoid yellowish lesion Figure A; by usiSurprisingly, the definitive diagnosis was indicative of an EH . Benign diseases are uncommon and, among these, EHs are a very entity with the largest series accounted for 43 EH cases.2 At careful revision of the pertinent literature, there aren’t reported specific radiological features of EH; generally, they appear as ovoid low-density endobronchial lesions,3 with a smooth edge, focal fat, or fat alternating with calcific foci .4 Such radiological findings appear substantially similar to those which may be found in BC tumors,5 this making the differential diagnosis based on radiological features only extremely challenging, as in the present case.Pure endobronchial neoplasm  represents a rare entity with a histological diversity.2 On the contrary, some authors6 suggested the surgical approach as first treatment, considering that an endoscopic biopsy need to be weighed against bleeding risk especially when a BC is suspected and the lesion is located, as the present case, at the level of main bronchus.Treatment of the benign endobronchial tumor is almost debated; surgical approach could be considered only in very selected cases that are difficult to diagnose or remove by a bronchoscopic approach.7Actually, in our opinion, the risk of bleeding in such cases could be remarkably minimized by just performing rigid bronchoscopy (instead of fiberoptic bronchoscopy). Moreover, as the present case clearly shows, a preliminary endoscopic removal may be extremely useful in all endobronchial lesions not only to solve the obstruction of the tracheobronchial tree but also to achieve a definitive diagnosis; this sometimes allows us to avoid unnecessary surgical resections for benign disease.5In our case, the radiological features were indicative for a BC; the endobronchial lesion appeared as a round-shape lesion with slightly lobulated borders and presented punctate calcifications that may be seen in 39% of centrally located bronchial tumors.8 A DOTA-peptide PET scan could have been a very useful non-invasive diagnostic method. Indeed, considering the high accuracy in detecting BCs,9 a negative result at DOTA-peptide PET scan would be highly disproving a diagnosis of BC. Therefore, when feasible, such imaging tools should be considered in the diagnostic work-up examination of suspected BCs.Moreover, the radiometabolic pattern (mild activity at 18-F FDG PET/CT scan) was also compatible with a well-differentiated BC tumor , as reported in several studies.In conclusion, hamartoma may rarely occur as endobronchial tumor with a pattern of clinical symptoms and radiological/radiometabolic features that may mimic an endobronchial carcinoid tumor. Thus, a preliminary endoscopic approach may be extremely useful not only to solve the obstruction of the tracheobronchial tree but also to achieve a definitive diagnosis; this sometimes allows us to avoid unnecessary surgical resections.


Although sunitinib is associated with a variety of adverse events, cases of sunitinib-related acute cholecystitis have rarely been reported. We herein report two cases of sunitinib-related acute acalculous cholecystitis in patients with clear cell renal cell carcinoma. In both cases, the gallbladder was surgically removed because it was difficult to improve the patient’s condition with the cessation of sunitinib and non-surgical treatment only. Attention must be paid to the possibility of sunitinib-related acute cholecystitis, which, although uncommon, can be life-threatening. After confirming the histological type of the tumor to be clear cell RCC based on a percutaneous kidney biopsy, sunitinib therapy  was started. During the 2 weeks of the first cycle, the patient experienced general fatigue, although no fever, right upper quadrant pain of Murphy’s sign were observed. Laboratory tests revealed elevated levels of C-reactive protein, lactate dehydrogenase, liver transaminases, alkaline phosphatase and amylase, while the white blood cell count and total bilirubin level were normal. After discontinuing the sunitinib therapy, the patient’s condition and laboratory tests improved. Two weeks later, he was readmitted for treatment with axitinib therapy and CT to evaluate the presence of pretherapeutic lesions. Despite exhibiting a normal gallbladder before sunitinib treatment, abdominal CT showed a tense and dilated gallbladder with surrounding fluid collection, but no gallbladder stones or emphysematous changes , and theA 67-year-old Japanese female with a right renal mass detected on an examination for the cause of gross hematuria was referred to our hospital. CT and magnetic resonance imaging (MRI) showed a hypervascular and inhomogeneous tumor in the right kidney with the duodenum over Gerota’s fascia and right renal vein invasion with distant metastasis to the lungs, thus suggesting a diagnosis of right RCC of clinical T4N0M1. After confirming the histological type of the tumor to be clear cell RCC based on a percutaneous kidney biopsy, sunitinib therapy  was started. The patient developed a fever during the 2 weeks of the first cycle; however, she exhibited no right upper quadrant pain, and Murphy’s sign was negative. Laboratory tests revealed only an elevated percentage of neutrophils, while the white blood cell count was within the normal limits. She was diagnosed with a bacterial infection, which was treated with broad-spectrum antibiotics, and the sunitinib therapy was discontinued. Three days later, laboratory tests disclosed elevated levels of C-reactive protein, total bilirubin, lactate dehydrogenase, liver transaminases, alkaline phosphatase and amylase, although the white blood cell count remained normal. Despite having a normal gallbladder prior to treatment, abdominal ultrasonography revealed thickening of the gallbladder wall without gallbladder stones or emphysematous changes ; therefoWe experienced two patients who underwent cholecystectomy to control acalculous cholecystitis caused by the administration of sunitinib. Although sunitinib is associated with a variety of adverse events, cases of sunitinib-related acute cholecystitis have rarely been reported. In the present two cases, it was necessary for the patients to undergo cholecystectomy in order to control acute cholecystitis.In the previous literature, only three cases of sunitinib-related acute cholecystitis have been reported, including one patient with a GIST  and two As sunitinib is widely used in the treatment of RCC and GISTs, the oncology community should be alerted to be aware of this uncommon and life-threatening adverse event.We herein described two cases of sunitinib-related acute acalculous cholecystitis in patients with clear cell RCC who ultimately underwent cholecystectomy to control their symptoms. Attention must be paid to the potential for sunitinib-related acute cholecystitis, which, although uncommon, can be life-threatening.


In 1942, Stout described tumors which derive from Zimmerman’s pericytes and suggested the term hemangiopericytoma (HPC). These tumors, which are often highly vascularized, pose difficulties in the surgical management regarding blood loss and complete resection. Therefore, preoperative management seems to be an essential part in dealing with these issues.We present a 70-year-old female patient with a large HPC in the pelvis. Preoperative embolization of the tumor was performed, and 2 weeks after the intervention, we completely resected the tumor with minimal blood loss.In which cases do we need preoperative treatment, especially emboliziation of hemangiopericytomas/solid fibrous tumors (SFT)? Although preoperative embolizations of tumors are now commonly undertaken, as for now, neither a clear statement nor a standardized approach has been given or developed. The purpose of this article is to provide our experience with preoperative embolization and to start a new discussion concerning a standardized approach. Hemangiopericytomas are tumors which derive from Zimmerman’s pericytes. Stout provided the first report and established the term hemangiopericytoma (HPC) . These aIn 1976, Smith et al. described the first case of preoperative embolization of retroperitoneal hemangiopericytomas . AlthougThese cases show that in highly vascularized tumors, especially in HPCs, preoperative arteographic imaging and subsequent embolization, even if only performed partly, reduce preoperative blood loss and increase the chance to perform a radical resection.The purpose of this article is to begin a new discussion concerning the approach to highly vascularized tumors, especially hemangiopericytomas. As to this date, a standardized approach, which takes these aforementioned aspects into account, does not exist.A 70-year-old female patient was presented with lower abdominal pain and constipation at our hospital. A computed tomography (CT) was performed and revealed a highly vascularized solid mass measuring 9 × 14 × 19 cm, congestion of the kidneys, and compression of the rectum  and a mass at the roof of the bladder (3 cm) as a sign of local recurrence. Figure Hemangiopericytomas are highly vascularized large tumors, which arise from pericytes, and are found more commonly in the abdomen or pelvis –12. TherDo we change the rate of recurrent disease if we perform a transection of these large tumors, as done in this case? Although, data presented by Espat et al. in 2002 showed with a collective consisting of 25 patients with a follow-up of 49 months metastases in 20 % and local recurrence in 4 % of the resected patients, interestingly, our patient did recur locally and distantly [In 2015, Manatakis et al. described a case of hepatic metastases of a HPC. In their final statement, they proposed transarterial embolization due to the “hypervascularity of the tumor”. If the case is read carefully, the authors describe a first liver resection without (!) embolization with an uneventful course and state that before the second liver resection, the transarterial embolization was used as tool to “bridge” . Also inBokshan et al. showed that even larger resections could be safely performed if in the hands of a dedicated center. In 2012, they published a case of a hepatic metastasized HPC and an ex vivo resection with veno-venous bypass, and reconstruction of the left liver vein with replacement of the inferior vena cava was performed without mortality .Table Due to the low-incidence of HPCs in the abdomen, a clear statement regarding a preoperative embolization  cannot be given. We propose following approach for tumors affecting visceral organs in the abdominal cavity:If the tumor is located in the abdominal cavity/pelvis and is large , an embolization needs to be performed. On the other hand, if the location and the size of the tumor are favorable, a hepatic resection can be performed straightaway.Certainly, the most crucial part of the treatment is a thorough discussion in the MDT.In our opinion, preoperative embolization should be incorporated into this interdisciplinary approach.Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.


A woman in her mid-60s presented with a bulky mass on the anterior abdominal wall. She had a previous incidental diagnosis of endometrial adenocarcinoma FIGO stage IB following a vaginal hysterectomy. Physical exam and imaging revealed a well circumscribed bulging tumour at the umbilical region, measuring 10 × 9 × 9 cm, with overlying intact skin and subcutaneous tissue. Surgical resection was undertaken, and histological examination showed features of endometrial carcinoma. She began chemotherapy and is alive with no signs of recurrent disease one year after surgery. This case brings up to light an atypical location of a solitary metastasis of endometrial carcinoma. Uterine cancer is the most frequently diagnosed gynaecological malignancy in developed countries. In 2008, more than 288,000 new cases were registered in the world, with a mortality rate of 2.2 per 100,000 women [The majority of women with endometrial carcinoma are diagnosed with localized early stage disease, yielding a high survival rate. The first step of treatment is complete surgical staging that can be followed by adjuvant radiotherapy and chemotherapy or both. In early stage disease, adjuvant radiotherapy has shown to significantly reduce locoregional recurrence, although it does not appear to increase overall survival or distant recurrence rates. Adjuvant chemotherapy can be considered in high-risk patients, but compared to radiotherapy alone, no differences were found in overall survival and progression-free survival, although further studies are needed. Combined modality treatment in high-risk patients was associated with a reduction in risk of recurrence or death in two randomized clinical trials and is the subject of the ongoing PORTEC 3 study . For medFollowing primary treatment, the overall recurrence risk ranges from 8 to 19% . TreatmeThe authors describe a case of an atypical location of endometrial carcinoma metastasis on the anterior abdominal wall without other signs of advanced disease.2) presented 3 years before with stage IV uterine prolapse and occasional vaginal spotting related to ulceration of the protruding cervix. Preoperative evaluation included a pelvic ultrasound that showed an enlarged uterus (L 89 × AP 58 × W 61 mm) with various small fibroids (around 20 mm) that distorted endometrial cavity hindering an accurate measurement of endometrial thickness. Both ovaries were normal in shape and size, and no malignancy was suspected. The optimization of the underlying diseases delayed surgery for several months. In July 2012, a vaginal hysterectomy was performed. Intraoperatively, inspection of both ovaries was unsuspicious. The histopathology examination revealed an enlarged uterus distorted by the presence of multiple fibroids and an intracavitary tumour invading more than half of the myometrium. The tumour was grade 3, poorly differentiated endometrial adenocarcinoma, without vascular space invasion, FIGO stage IB. Given her general condition and absence of signs or symptoms of the disease, despite incomplete surgical staging and high intermediate-risk disease, it was decided by a multidisciplinary team of gynecologic surgeons, pathologists, medical, and radiation oncologists to proceed with adjuvant radiotherapy. Chemotherapy was not considered due to her medical and social background. The patient received 50 Gy (25 fractions of 2 Gy) of external pelvic irradiation and 24 Gy (4 fractions of 6 Gy) of brachytherapy to the vaginal cuff.A Caucasian woman in her mid-60s, gravida 1 para 1, with toxic multinodular goiter with an intrathoracic extension, hypertension, chronic anaemia, and obesity , lactate dehydrogenase mildly raised (333 UI/L), and elevated CA 125 (478 UI/mL). Magnetic resonance imaging (MRI) Figures  revealedSurgical resection of the tumour was performed followed by peritoneal washing and bilateral salpingo-oophorectomy. Reconstruction of the abdominal wall was accomplished with the use of a synthetic mesh. The cross section of the tumour is shown in 2) and carboplatin (300 mg/m2) and completed six cycles. At the end of treatment, radiologic imaging with thoracic and abdominal CT and pelvic MRI did not show any signs of recurrence. More than one year after surgery, the patient is alive and asymptomatic.She began chemotherapy one month after surgery with paclitaxel , spleen (1%), central nervous system (<1%), extra-abdominal lymph nodes , and, more rarely, adrenals, pancreas, and appendix –13.In this case and taking into account its location near the umbilicus, the hypothesis of an umbilical metastasis, also termed Sister Mary Joseph's nodule, was raised. Sister Mary Joseph's nodule most typically presents as an irregular lump on the umbilicus, ranging in size from 0.5 to 2 cm, although there are reports with nodules reaching up to 10 cm. It may be ulcerated and necrotic and have a bloody, mucinous, serous, or purulent discharge. It can be detected before or during diagnosis of the primary tumour or after treatment. This umbilical metastasis is found in 1–3% of patients with gastrointestinal or genitourinary malignancy, including endometrial cancer , 14. TheAbdominal wall metastases have also been linked to surgical incision, regardless of the surgical approach (laparotomy or laparoscopy). Although rare, it has been described in cases of endometrial carcinoma –23. The Park and Hwang reported a case of a postmenopausal woman with an abdominal wall metastasis in stage IA serous endometrial adenocarcinoma, eight months after surgical staging. Treatment consisted of surgical excision and chemotherapy, and three years after surgery she is alive with no signs of disease . CompariThis case highlights an unusual location of a solitary metastasis of endometrial cancer on the soft tissue of the abdominal wall. Almost all cases reported in the literature are diagnosed after primary treatment and are related to the surgical incision. In this case, it is difficult to know exactly how neoplastic cells implanted in the soft tissue and developed into a mass. Surgical resection was feasible and the patient completed chemotherapy with no signs of recurrence after one year.


Internal hernia after gastrectomy is a rare complication. It can progress rapidly to vascular disturbance, necrosis, and perforation, therefore early diagnosis and surgical treatment is essential. We present a case of internal hernia following laparoscopic-assisted proximal gastrectomy with jejunal interposition reconstruction in a 68-year-old man, who presented with acute abdominal pain and vomiting. Computed tomography showed a whirl sign, ascites, and a closed-loop formation of the small intestine. We diagnosed an internal hernia and performed emergency surgery. Laparotomy revealed chyle-like ascites and extensive small intestine with poor color. We recognized that about 20 cm of jejunum from the ligament of Treitz was strangulated behind the pedicle of the jejunum lifted during laparoscopic-assisted proximal gastrectomy. We relieved the strangulation, whereupon the color of the strangulated intestine was restored. Therefore, we did not perform intestinal resection and reconstruction. Finally, we fixed the jejunal pedicle and mesentery of the transverse colon. We report this case as there are few reported cases of internal hernia after laparoscopic-assisted proximal gastrectomy. Laparoscopic gastrectomy for gastric cancer is one of the common treatments for patients with early gastric cancer, not only in Asia but also in Western countries, with numerous clinical series being reported –3. InterOne of the forms of internal hernia is Petersen’s hernia, which is an internal hernia that can occur through a mesenteric defect caused by Roux-en Y anastomosis (RY). Use of the RY reconstruction during distal and total gastrectomy has been gradually increasing in recent years, most likely to avoid anastomotic leakage and reflux inflammation , 5. FurtThis case of internal hernia was formed by passing behind the jejunal pedicle at the side of the laparoscopic-assisted proximal gastrectomy (LAPG). There are few reported cases of internal hernia after LAPG, so we report this case as our experience with internal hernia since the introduction of LAPG with retrocolic JI reconstruction.A 68-year-old man presented with sudden abdominal pain and vomiting. He had no other symptoms. He underwent LAPG for early gastric cancer 2 years earlier. Laboratory test results were almost normal, including those for tumor markers , cancer antigen (CA) 19-9)  show further benefits including reduced bleeding and improved quality of life when compared with a traditional open distal gastrectomy , 10, 11.However, there are several reconstruction methods for proximal gastrectomy, such as JI, EG, JPI, and DT. In our institute, we have performed JI reconstruction following LAPG with a retrocolic approach for a long time. For the surgical procedure of LAPG, the stomach was almost half transected. After the jejunum was divided about 20 cm from the ligament of Treitz, the distal jejunum was placed posterior to the transverse colon. An end-to-side anastomosis was performed by using one 25-mm circular stapler for the esophagojejunostomy, and one 60-mm linear stapler was used for the jejunal stamp. We designed the length of free jejunum to be about 12 cm; an end-to-end anastomosis was performed for gastrojejunostomy. Finally, a side-to-side jejunojejunostomy was performed. We consider JI reconstruction to be the most physiological anastomosis, because the distal stomach remains to maintain normal digestive function, and the JI reconstruction has a similar role to the cardiac sphincter in reducing gastroesophageal reflux inflammation , 9. JI rWe experienced a case that was diagnosed as internal hernia after LAPG with JI reconstruction for gastric cancer. Internal hernia is a rare complication which, however, needs to be diagnosed quickly and treated with emergency surgery.Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.


Candida albicans in our case. Antifungal therapy was started and the chest CT findings showed a remarkable improvement. To our knowledge, this is the first case report describing multiple pulmonary cavitary lesions in invasive candidiasis.We herein report a case of invasive candidiasis presenting rare findings on chest computed tomography (CT). The chest CT scan showed multiple small cavitary lesions and nodules with surrounding ground-glass opacity, and also bilateral pleural effusion. Although this CT finding is thought as specific for pulmonary aspergillosis, two sets of blood culture specimens were drawn which yielded  It has been reported that invasive candidiasis is rare but associated with considerable mortality in immunocompromised or critically ill patients. The most common chest computed tomography (CT) findings of invasive candidiasis have been reported as multiple bilateral nodules often associated with air-space consolidation . Here, wth day, he presented fever and hypoxemia. He had a history of stage IV chronic kidney disease secondary to hypertensive nephrosclerosis for 15 years, and distal gastrectomy for gastric cancer 20 years before. He did not have smoking history and risk factors for HIV infection, and drank alcoholic beverages occasionally. Physical examination revealed poor oral hygiene only. The chest x-ray revealed multiple nodules in the right upper lung field, and also mixed ground-glass and airspace opacities in the entire right lung  catheters. His small bowel obstruction did not resolve, and on the 8Candida spp. exist as normal flora of the human skin, oropharynx, lower gastrointestinal tract, and genitourinary system. Candida spp. are now one of the most common causes of nosocomial blood stream infections worldwide [Candida spp. enter the bloodstream. Once the fungus is in the bloodstream, it can spread to other parts of the body and cause infection. There are risk factors for invasive candidiasis, such as central venous catheter, surgical procedure, acute renal failure, disseminated intravascular coagulopathy, parenteral nutrition and the use of broad-spectrum antibiotics [orldwide . Invasivibiotics . In our Candida from blood, normally sterile organ or body cavity [Candida albicans, and this CT finding is rare in invasive candidiasis. It was reported that cavitary lesions were less common in invasive candidiasis (4%) than in aspergillosis (16%) [The few available studies on this topic indicate that pulmonary manifestations of invasive candidiasis are seen in no more than 0.2 to 8.0% of at-risk ICU patients and cancer patients [y cavity ,4. We cois (16%) , and, toIn conclusion, multiple pulmonary cavitary lesions are rare CT manifestations of invasive candidiasis. This CT finding is thought as specific for pulmonary aspergillosis, however, we should also consider this finding in invasive candidiasis occurring in patients who have risk factors. We believe that our case will be helpful to the understanding and recognition of the spectrum of this rare condition.Written informed consent was obtained from the patient for publication of this Case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.


Hepatocellular carcinoma (HCC) is a common cancer all over the world. It demonstrates a tendency for vascular invasion, producing extensive intrahepatic metastases and portal vein or inferior vena cava extension. Tumor spread of abdominal diseases via hepatic ligaments has also been reported. The author reports a rare case of HCC invading the anterior abdominal wall muscles and protruding into the subcutaneous fat. Hepatocellular carcinoma (HCC) is the most common primary tumor of the liver and the third leading cause of cancer related death , 2. Cirr32 year old gentleman was known to have Hepatitis B virus presented to our institution complaining of right upper quadrant pain, distension, jaundice and vomiting. Laboratory findings shows mildly elevated ALT/AST and highly elevated alpha fetoprotein levels reaching 1518 ng/ml.CT scan was performed and showed a large mass in the left hepatic lobe with area of central necrosis demonstrating peripheral isodensity to the liver and invading the greater momentum  with mulUltrasound guided biopsy of the liver lesion was performed and the patient developed sudden drop of hemoglobin following the procedure. CT scan performed and showed interval development of intralesional hemorrhage. After discussing the plan of palliative management with the patient, he refused to start chemotherapy and decide to be discharged against medical advise.HCC is responsible for about 1 million deaths annually worldwide. The difficulty in treatment of this cancer and the reason for high mortality is the association of this cancer with cirrhosis which limits both the treatment options and increases morbidity of any modality of treatment. HCC is usually asymptomatic at early stages and has a great propensity for intravascular invasion even when the tumor is small; hence HCC is usually at an advanced stage when discovered. The most common sites of metastases of HCC include lung, peritoneum and lymph nodes. In a study by Katyal et al., tabulation of all extra hepatic metastatic sites showed the most common sites to be the lung in 81 (55%) patients, abdominal lymph nodes in 60 (41%) patients, and bone in 41 (28%) patients . Rare siHCC is a highly malignant tumor that can invade and metastasize to almost any organ in the body. Therefore unusual areas of metastases should not disclose the radiologist from calling it as metastasis. Although tissue biopsy is needed in unusual cases.


We report a case in which an immature teratoma developed following three previous resections for mature cystic teratomas. The patient was a 26-year-old nulliparous woman with a regular menstrual cycle. Twelve years earlier, she had consulted a pediatrician for complaints of lower abdominal pain. Bilateral cystic teratomas were suspected and she underwent a left salpingo-oophorectomy and a right cystectomy laparoscopically, and bilateral mature cystic teratomas were diagnosed histologically. She underwent a right cystectomy twice afterwards and mature cystic teratomas were diagnosed. Three years after the third surgery, a regular checkup performed annually for ovarian cyst recurrence revealed a 9.3 cm ovarian cyst by ultrasonography without marker elevation or complaint of symptoms. Magnetic resonance imaging (MRI) showed a 10 cm multilocular cyst, including a part with heterogeneous medium and high-signal intensity on T2-weighted images, which revealed enhancement on dynamic contrast-enhanced MRI unlike the previous images. Ovarian tumors, including immature teratomas and malignancy, were considered. She had a strong wish to undergo laparoscopic surgery. She was diagnosed with an immature teratoma, grade 1 of the right ovary. Although the frequency of recurrence of immature teratomas after resection of mature cystic teratomas is very low, regular checkups are necessary because there may be no associated symptoms. Teratomas are the most common type of ovarian germ cell neoplasm, comprising approximately 15% of all ovarian tumors. They occur within a very wide age range but usually occur during the reproductive years. Mature cystic teratomas are rarely large  and often bilateral 15%–25%) [5% [2]. MThe patient was a 26-year-old nulliparous woman with a regular menstrual cycle. Twelve years earlier she had consulted a pediatrician for complaints of lower abdominal pain. Ultrasonography revealed a giant pelvic cyst, mostly echogenic, which was larger than fist size. Carbohydrate antigen 19-9 (CA19-9) and squamous cell carcinoma antigen (SCC) were elevated to 169 U/mL and 16 ng/mL, respectively. Magnetic resonance imaging (MRI) showed a large 12 cm unilocular tumor on the left ovary and a 4 cm tumor on the right ovary with high-signal intensity on T1-weighted images and signal dropout on fat suppression imaging. Bilateral cystic teratomas were suspected and she underwent a left salpingo-oophorectomy and a right cystectomy laparoscopically, and bilateral mature cystic teratomas were diagnosed histologically.CA19-9 and SCC values were in the normal range after tumor resection. Eight years after the first surgery, a regular annual checkup for ovarian cyst recurrence revealed swelling of the right ovary by ultrasonography, without elevated CA19-9 and SCC. The patient had no complaints. MRI showed a 6.5 cm multilocular ovarian cyst with signal decrease on fat-saturated T1-weighted images and these findings were consistent with recurrence of cystic teratoma . She undImmature teratomas are uncommon (1% of ovarian teratomas) and affect younger women , 7. TherThis brief report presents the case of a patient with an immature teratoma that developed after three previous surgeries for mature cystic teratomas. Although the frequency of recurrence is very low, regular checkups are necessary because there may be no associated symptoms.


Tuberculosis remains a worldwide health problem causing morbidity and mortality. Abdominal tuberculosis is a rare form of the disease. Abdominal form of tuberculosis can mimic other non-infectious diseases. In this report, we presented an abdominal tuberculosis presenting with an intra-abdominal mass lesion and multiple lymphadenopathies that mimics lymphoma. Tuberculosis (TB) remains a worldwide health problem causing morbidity and mortality particularly in developing and low-income countries. Abdominal TB is a rare form of the disease and also diagnosis may be challenging due to non-specific signs and symptoms of the disease. Abdominal form of TB can mimic other non-infectious diseases such as; inflammatory bowel disease, lymphoma and peritoneal carcinomatosis. Herein, we present a case report with abdominal TB presenting with an intra-abdominal mass lesion and multiple lymphadenopathies that mimics lymphoma.A 57-year-old male presented with abdominal pain, fatigue, high fever, and weight loss of 6 kg for two months. His previous medical history was unremarkable. Physical examination revealed pain on the left side of the abdomen and hepatosplenomegaly. Laboratory findings were as follows: erythrocyte sedimentation rate 86 mm/h, leukocyte 12600/µl, neutrophile 7200/µl, thrombocyte 510000/µl, monocyte 2100/µL, hemoglobin 10.2 g/dl, hematocrit 30%, MCV 74.6 fl, and lymphocyte 2200/µl. Computed tomography (CT) of the chest showed multiple mediastinal lymphadenopathies (maximum size 15 mm) and bilateral apical fibrotic bands. A uniform-density mass lesion of 70 mm that filled retrovesical and left pelvic area was detected in abdominal CT. The lesion surrounded superior part of sigmoid colon and there were multiple lymph nodes in the retro-vesical, celiac, para-aortic and para-iliac areas . HistoloAbdominal TB rarely presents with abdominal mass, so distinguishing from a malignancy is not easy. In this case, abdominal CT findings suggested a malignancy, especially lymphoma. A few TB cases were reported to present with findings mimicking cervical carcinoma , over caAbdominal TB is a rare form of tuberculosis. It can present with symptoms and signs mimicking lymphoma. It should be taken into consideration that TB can present like a malignant abdominal mass in countries with a higher TB prevalence, especially in the developing countries, by physicians and radiologists.


Abernethy malformation is a rare congenital vascular abnormality in which the portal vein bypasses the liver and drains directly into the inferior vena cava. Diagnosis is complex and requires good quality imaging methods to identify details in systemic and portal circulation in order to establish diagnostic confirmation and treatment strategy. In this study we highlight the significance of the use of CT scans and Color Doppler Duplex Ultrasound for the diagnosis, treatment and evolution assessment in two adults with Abernethy malformation.The diagnosis and the treatment of two patients with Abernethy malformation by CT scan and Color Doppler Duplex Ultrasound is described. One patient was submitted to liver transplantation due to chronic liver disease and multiple nodules diagnosed as adenoma. The other patient had normal liver function and a mild neurological and psychomotor dysfunction, therefore we adopted clinical treatment and close liver parenchyma evaluation and nodule surveillance, using an imaging approach involving intercalating CT scan and Color Doppler Duplex Ultrasound every 6 months. We highlight some important direct and indirect findings of non-invasive imaging methods.Abernethy malformation requires meticulous image diagnosis to improve treatment and avoid iatrogenic procedures. CT scans and Color Doppler Duplex Ultrasound are both efficient methods for diagnosis, treatment planning and evolution assessment of patients with Abernethy malformation. Abernethy malformation (AM) is a rare congenital abnormality described by John Abernethy in 1793. It is characterized by a shunt between the portal vein and systemic circulation . It is fThere are two types of AM demonstrated on Fig. A meticulous assessment of the liver and vascular anatomy by non-interventional abdominal exams in Abernethy malformation aids diagnosis and planning of appropriate treatment. In this study, we detailed some important direct and indirect findings of non-invasive imaging methods and highlight the value of CT scans and Color Doppler Duplex Ultrasound for the diagnosis, treatment and evolution assessment of two adult patients with Abernethy malformation.The first patient was a 20-years-old female with chronic renal failure and systemic arterial hypertension, who had been on dialysis since February 2012 and on the waiting list for kidney transplantation. She had heart surgery at the age of 8 to treat a congenital heart disease (CIA type ostium secundum) and a disabled factor XII of 20 % (thrombotic factors predisposes). She also presented delayed psychomotor and learning development. Clinical examination showed no external phenotypic malformation evidence. In genetic assessment, we found a heterozygous A1298C MTHFR gene mutation. During abdominal investigation for renal transplantation, an ultrasonography and CT scan detected chronic liver disease, multiple nodules and the portal vein with a regular splenomesenteric junction path, but draining directly into the inferior vena cava, close to the right atrium as described in the pathology of Type 1 Abernethy malformation, demonstrated in Fig. In the second case, the patient was a 52-year-old female with celiac disease and hypothyroidism, diagnosed 10 years ago. Initially asymptomatic, AM was first suspected after a routine ultrasound exam six years ago for abdominal pain, at the time diagnosed as gastritis. The angio CT Fig.  clearly This study was approved by the Institutional Review Board fulfilling all requirements for studies in humans, following the guidelines of the 1975 Declaration of Helsinki.AM is an extremely rare syndrome, with less than 20 cases reported in English language medical literature. Most patients with AM suffer premature mortality as a result of the serious conditions related to this syndrome. In those patients with mild conditions, the presence of a portocaval systemic shunt is often not recognized during childhood . This diThe second case was initially presumed to be agenesis of the portal vein with decompensated hepatic dysfunction. However, after sagittal and coronal scans beyond the axial planes of the abdominal multidetector tomography, and detailed Doppler Duplex liver ultrasonography exams, the abnormal liver vascular anatomy was identified as AM. Occasionally, the image diagnosis is easy to make , but in Portal vein thrombosis exclusion is also a differential diagnosis of AM. Currently, cavernous transformation of chronic portal thrombosis can be identified with high accuracy using Doppler Duplex Ultrasonography exams and CT imaging . The diand, 3rd and 4th branch portal ramification, usually visualized in the conventional imaging methods), as the enlarged and hypertrophied hepatic artery in the hepatic hilum.The combination of both imaging methods, first US and than CT gives a better chance of detecting rare liver disease, considering also, a recently revision that suggests the US as the first line option to assess liver disease . We showThe main advantages of US are that it is non-invasive, available in most health centers, and is not expensive . HoweverThe association of portal vein anomaly and nodular liver lesions was observed in almost half of the reported cases, and the most frequent lesion is the focal nodular hyperplasia . The treIn summary, we reported and detailed some important direct and indirect findings of non-invasive imaging methods. On two cases of AM in an attempt to evaluate the significance of CT angiography and Duplex Doppler liver plane anatomy to identify variations of liver anatomy and vascular malformations. These imaging methods are crucial to improve the clinical diagnosis of AM and to avoid iatrogenic procedures when planning liver surgery strategy.Written informed consent was obtained from both patients for publication of this Case report and any accompanying images. A copy of the written consent is available for review by the Editor of this journal.


Elephantiasis of the external genitalia is characterized by lymphedema and thickening of the subcutaneous tissues. This gives the skin an appearance similar to a pachyderm skin. This pathology is invalidating for the patient. Reconstructive surgery is often the only way to restaure aesthetic and functional aspects of the external genitalia. We aim to report a 52 year man with huge penoscrotal elephantiasis who underwent excision and penoscrotal reconstruction at the department of Urology, Yalgado Ouedraogo Teaching Hospital at Ouagadougou. Penoscrotal elephantiasis can lead to hudge enlargement of the scrotal sac and the penis. It is as a result of lymphatic obstruction and subsequent infiltration of the subcutaneous tissue of the external genitalia with lymph . The cauWe aim to report a case of penoscrotal elephantiasis which was managed at the Department of Urology, Yalgado Ouedraogo Teaching Hospital at Ouagadougou (Burkina Faso).A 52 year old cultivator, presented to the department of Urology, Yalgado Ouedraogo Teaching Hospital with history of gradual enlargement scrotal sac over twelve years. The swelling was painless and itchy. No history of a lower urinary tract symptoms, pelvic surgery or venereal disease. Physical examination revealed a hudge scrotal sac extending down to the upper third of the legs Figure . The scrAnterior midline incision was made over the scrotal mass, this delivered the penis. The skin of the inner side of the tunnel was found to be normal and was preserve to cover the penis Figure .Looking at the epidemiological and clinical findings, we made a diagnosis of penoscrotal elephantiasis. The patient had taken a dose of anti filarial drugs . He washed the scrotal mass twice per day for three days with a polividon solution. Preoperative testing was performed before surgical procedure. He underwent surgical excision of the subcutaneous tissues and reconstruction of his penis and his scrotum. The procedure was performed as follows:Two oblique incisions were made toward the groin starting from cranial end of the previous incision, to find and dissect the spermatid cords and testicles.Excision of subcutaneous tissue with careful haemostasis.Resection of all abnormal tissue covering the penis.Fixation of the testicles to the bottom of the skin flaps to be used for scrotal reconstruction.Excision of excess skin.Drainage of both hemi-scrotum which were closed separately using absorbable sutures Figure .Bladder was drained with size 20 F foley’s urethral catheter.The excised scrotal mass contained gelatinous liquid. The subcutaneous tissue was very thick and whitish. All resected tissue weighed 12 kg.The postoperative outcomes were marked by anaemia, which was corrected by compatible blood transfusion. The drains were removed on the fourth post-operative day. No further complication was noted. The penis and the scrotum were cosmetically acceptable at 3 weeks and 5 months post-operative review. The scrotal sac was much reduced and flexible Figure .Figure 4The penoscrotal elephantiasis can be defined as massive enlargement of the scrotal sac secondary to a subcutaneous accumulation of lymphatic fluid. It is also called lymphedema , 2. The In tropical countries, microfilaria is the leading cause of elephantiasis of the male external genitalia . The diaPrimary or congenital elephantiasis. In these cases, there is a congenital abnormality of the lymphatic system leading to lymphatics obstruction. It may manifest early or later (Meige disease) in life.Secondary elephantiasis. These may be caused by parasitic infection (lymphatic filariasis), bacterial infection, urethral stenosis, pelvic tumor or radiotherapy –10. TreaConserving surgery: this is a lymphangioplasty in order to improve lymphatic drainage. These techniques have been abandoned due contradicting results .Surgical excision: its principle is complete removal of all affected tissue  and penoscrotoplasty. Reconstructive techniques vary , 4, 5. MThough the diagnosis is made clinically, some laboratory test may be of importance. The microfilariae may be found in the serum, ultrasonography of scrotal sac may pick the adult worms and isotope bi pedal lymphography or radiography may be needed to outline the lymphatics . The aetExternal genitalia elephantiasis is a debilitating condition. It causes both psychological and physically distress. When it affects the external genitalia, there may delay in presentation due to private nature of the area. The commonest aetiological factor in the tropics is microfilariasis. Surgical excision followed by a penoscrotoplasty gives excellent result.Written consent was obtained from the patient for publication of this Case report and any accompanying images.