Although the incidence of gastric cancer has decreased worldwide, it is still among the most common cancers worldwide. Usual manifestations of gastric cancer include gastrointestinal-related symptoms such as weight loss and abdominal pain. Isolated symptomatic bony metastasis at initial presentation is very rare. Here, we present an infrequent case of a middle-aged lady who presented with a left femur neck fracture and left eye esotropia as the initial presenting clinical manifestations of gastric cancer. She was found to have an isolated bone metastasis seen throughout the body skeleton with no extraosseous metastasis. The patient was treated conservatively and received external beam radiation therapy as part of palliative therapy. This report signifies the importance of considering the diagnosis of gastric cancer in middle-aged patients with isolated symptomatic bone metastasis. It also reviews the overall poor prognosis of such cases and recommends further studies on the role of chemotherapy and radiation therapy in such cases. Gastric cancer is among the most common cancers worldwide, and it was the fourth most common cause of cancer deaths in 2020. According to World Health Organization (WHO), a total of 1.09 million new cases and 769,000 deaths were recorded in 2020 . HoweverA 54-year-old female patient was referred to our hospital with chronic pain in the left hip for two months, associated with generalized body ache and easy fatigability. She reported that her left hip pain was getting worse over time without a preceding trauma and is aggravated by physical activities. She had no nausea, vomiting, hematochezia, melena, jaundice, fever, night sweats, or abdominal pain. Her past medical history was significant for type II diabetes only, and her family history was negative for any malignancies. The patient was admitted for further evaluation and workup. The initial laboratory workup results are illustrated in Table Upon admission, her left hip pain became intolerable to the point where she could not bear her weight or ambulate alone. On bedside examination, she was found to have a left eye medial squint on general inspection. Her abdomen was soft, not tender, and not distended. Musculoskeletal examination showed shortened and externally rotated left leg. The range of motion in the left hip was extremely limited, and the patient could not abduct or flex her hip. There was tenderness all over the left hip and thigh, but no deformities were noted, and pulses were intact. A plain X-ray of the pelvis revealed a pathological fracture of the left femur neck with incidental multiple lytic lesions involving the whole pelvic bones as seen in Figure As the initial presentation included left eye esotropia, MRI of the brain and spine were done to rule out brain metastasis. MRI showed central skull base mass with multilevel bone marrow replacing lesions in the cervical and thoracic spine  as shown in Figure During her hospital stay, she developed anorexia and multiple episodes of vomiting. The occurrence of those symptoms along with the elevated cancer antigen 19-9 (CA19-9) levels found in the serum at the initial laboratory workup prompted a workup for gastrointestinal malignancy. CT of the chest, abdomen, and pelvis was done, which was unremarkable. Upper GI endoscopy was done and showed a 2 cm ulcerated lesion at the gastric antrum. Multiple biopsies were taken, and the pathology report showed poorly differentiated, diffuse type, signet-ring gastric adenocarcinoma.The management plan included limb traction, ambulation restriction, and analgesics as needed as part of conservative treatment. Surgical intervention was not an appropriate decision with her extensive disease condition. The patient was prone to multiple other iatrogenic fractures with any surgical intervention including total femur replacement. She eventually received external beam radiation therapy with 08 gray single-shot to skull base metastasis for abducent nerve compression as part of palliative therapy. As there was no rule for any surgical management for her advanced gastric cancer, she was referred to the medical oncology department for further palliative chemotherapy.Although the incidence of gastric cancer has decreased worldwide, it is still the fourth most common cause of cancer deaths in the world as reported by WHO statistics in 2020 . GeneralMost of the patients present with GI-related symptoms at initial presentation, which include abdominal pain, weight loss, dysphagia, nausea, vomiting, early satiety, and anemia ,9. It isAlthough 85% of reported cases of metastatic gastric cancer to bone also metastasized to visceral organs ,11, withIn the differential diagnosis of such presentation, it is important to include multiple myeloma as differential diagnosis as pattern mimics its clinical manifestation ,16. HowePatients presenting with bone metastasis are considered to have a poor prognosis. The role of chemotherapy and radiation therapy is still unclear in such cases. Kim et al. performed a cohort study on 1,342 patients with metastatic or recurrent gastric cancer, the initial bone metastasis rate was 6.7%, and the median survival age was 4.4 months after the diagnosis of bone metastasis. In their study, the results supported the conclusion of poor prognosis of such cases; however, the patients who received chemotherapy had a better prognosis as compared to those who did not .In conclusion, our case report here demonstrates an unusual presentation of gastric adenocarcinoma, which is manifested with isolated symptomatic bone metastasis. Cases presenting with similar characteristics are rarely documented in the literature. The current case report highlights the importance of considering metastatic gastric cancer as a differential diagnosis in patients presenting with pathological fractures and left abducent nerve paralysis as a symptom of extensive bone metastasis. Our review of the literature concluded that similar cases generally have a poor prognosis. Although symptoms can be treated conservatively, no definitive treatment can be offered at this terminal stage. The role of chemotherapy and radiotherapy in such patients is unclear and needs further research.


Prostatic cysts are rare, usually asymptomatic and detected incidentally at imaging. Midline prostatic cysts are less common and mostly located posteriorly. We describe a case of a 51-year-old man with unknown comorbidities who presented with troublesome irritative and voiding symptoms. Transabdominal and transrectal ultrasound, abdominal computed tomography and pelvic magnetic resonance imaging revealed a midline intraprostatic cyst protruding into the bladder lumen, with no communication with the urethra on voiding cystourethrography. Uroflowmetry findings suggested an obstructed flow. The patient underwent transurethral marsupialization of the cyst, which was found to project on the bladder outlet causing urinary obstruction, with excellent outcomes. What makes this case further interesting is the unusual cyst’s relationship with the patient’s prostate and bladder neck. To our best knowledge, this is so far the seventh reported case in the literature to describe an anteriorly placed midline intraprostatic cyst projecting at the bladder neck region and causing bothersome lower urinary tract symptoms. Prostatic cysts are uncommon, observed in 0.5 to 7.9% of patients. They have mostly an embryological origin or both cause-and-effect relationship with chronic prostatitis , 2. BesiA 51-year-old man presented with increased urinary urgency, frequency, hesitancy, poor intermittent stream and dysuria of 15 months’ duration. He denied any history of pelvic or perineal trauma, urethral instrumentation, urological disease or neurological deficit. He described his LUTS as moderate and stated that he has been extremely bothered over the last few months. He had three children and did not have any complaint of gross hematuria, urethral discharge, erectile dysfunction, haematospermia or post-coital discomfort, other ejaculatory disorders or infertility.Digital rectal examination revealed a smooth, normally firm, and non-tender prostate gland. Urinalysis did not show microscopic hematuria, culture was sterile, and cytology was negative for malignancy. The serum-specific prostatic antigen level was 0.51 ng/mL and routine blood tests were within normal limits. Uroflowmetry revealed a maximal flow rate of 10 mL/s with a flat curve, voided volume of 290 mL, voiding time of more than 2 minutes, and the postvoid residual (PVR) urine volume was 90 ml, suggesting an obstructed flow.Abdominal and transrectal ultrasonography (TRUS), and abdominal computed tomography (CT) showed an isolated, anteriorly positioned and midline intraprostatic cystic lesion with an approximate diameter of 1.5 cm that projected at the level of the bladder neck region Figure .In order to investigate further, pelvis magnetic resonance imaging (MRI) revealed an isolated anteriorly placed MPC of 1.7 x 1.5 x 1.3 cm arising from the upper part of the bladder neck region and protruding into the bladder lumen, with no evidence of any communication with the urethra , transurethral and percutaneous unroofing or marsupialization of the cyst using wire loop, Collin’s knife or Holmium laser, and less frequently, open surgery -9, 15. TOur intraoperative observation of incomplete marsupialization of the cyst, which was located in a challenging position to be safely unroofed via a transurethral route, supports Al-Nasser and Almannie's recommendation , 15. AccMPC is an uncommon condition and mostly asymptomatic, yet it should be considered as a differential diagnosis in men with LUTS. Anteriorly placed MPCs are extremely rare, but their surgical management may be technically challenging and harbor the potential risk of retrograde ejaculation as most cases arise from the bladder neck region. Hence, an individual diagnosed with such a condition should undergo a focused diagnostic workup to establish the cause-and-effect relationship with the patient’s complaints. When symptomatic, the cyst should be managed conservatively. Transurethral marsupialization of the cyst can be used in selected cases , whereas, the percutaneous approach could be superior to transurethral techniques, providing efficient, more secure, and effective treatment option to unroofing an anteriorly placed MPCs protruding into the bladder lumen, especially when approaching symptomatic and sexually active young patients wishing to preserve ejaculation and fertility.


Ovarian masses are not easily differentiated on transabdominal ultrasound in children. A useful supplement in various pediatric applications is dynamic contrast-enhanced ultrasound (dynCEUS). It can be performed quickly and easily. However, the literature for dynCEUS on pediatric ovarian masses is limited. We compared two cases with ovarian teratoma in which dynCEUS was a helpful additional tool. Non-specific chronic to acute abdominal pain is the primary clinical symptom of ovarian masses that often lead to a visit to the emergency department. Transabdominal B-mode ultrasound (US) is then the initial imaging modality. Neoplasms of the ovaries in girls are rare, with an incidence of only 3 cases per 100,000 per year . In almoAdditional magnetic resonance imaging (MRI) is helpful in differentiation due to the advantageous high contrast resolution for soft tissue. Therefore, MRI is considered the gold standard of modern diagnostics without ionizing radiation in the evaluation of pelvic masses . Diffusi®) distribution is possible during the examination. The intravenously applied microbubbles remain strictly intravascular and the gating of reflex microbubbles represents the vascularization and perfusion of a mass (Time-resolved dynamic contrast-enhanced ultrasound (dynCEUS) is another diagnostic modality. Lately, the use of dynCEUS has been increasing significantly in pediatric cases. Dynamic CEUS is quick and easy to perform in pediatric patients and is very well-tolerated . Visual f a mass . Therefof a mass , 10. In f a mass . These t® for intravenous application in pediatric liver imaging. The recommended dosage depends on the weight of the child. The dosage is 0.03 ml per kg bodyweight with a maximum limit of 2.4 ml per injection  intravenously per year of life, followed by a 10 ml bolus of NaCl-solution.Scans were performed with a Siemens  ultrasound machine by an experienced pediatric radiologist trained in CEUS with a 6-1 MHz curvilinear transducer in both cases using the same abdominal preset. In childhood and adolescence, the ultrasound contrast media is applied depending on age (0.1 ml/year of life) . Recentlnjection . For conOne cycle of contrast was sufficient to characterize the lesion. For accurate quantitative evaluation of perfusion dynamics, a video clip of ~120 s was recorded and stored. For software evaluation, the position of the transducer was not allowed to change with the onset of contrast media injection.®, Bracco Imaging, Germany) was used  in serum was slightly elevated at 15 ng/ml. The other tumor markers and laboratory values were unremarkable. B-mode ultrasound and a multimodal MRI with DWI and contrast media visualized a cystic solid mass of a maximum of 15.5 cm depending on the left ovary. A contained irregularly configured solid portion without calcifications was ~7.0 cm in size and with vigorous perfusion on Doppler ultrasonography . AnalysiBased on the two different pediatric cases with an ovarian mass presented here, it can be shown that dynamic CEUS with subsequent quantitative analysis is a new diagnostic method that appears to be able to better differentiate lesions. An ovarian mass in girls rarely occurs and differentiation of this lesion is sometimes not straightforward . In thisOf particular importance in all diagnostic procedures is that vascularization is assessed, as it correlates with the invasive potential of a lesion . Thus, aIn contrast dynamic CEUS can visualize vascularization and perfusion in real-time using intravascular microbubbles. The echogenicity of the bubbles in a tissue corresponds to the volume of blood contained therein . AccordiStudies in adult women have already demonstrated the value of dynamic contrast-enhanced ultrasound in malignant ovarian tumors , 10. MalTo our knowledge, except from a single case reports , the diaFor example, Madenci et al. showed that girls who had in imaging a simple cyst in the ovary never associated it with malignancy . PredomiOur case of an immature teratoma with potential malignant changes is in agreement with all these findings in adults and children. The tumor was very large, 16 cm in diameter. Dynamic CEUS and quantitative functional analysis showed significantly higher relative values for PE, RT, WiAUC, and WiWoAUC in the suspicious solid part compared to the first case of a mature teratoma. It must be remembered that the calculated absolute values of PE, WiAUC, and WiWaAUC are not well-comparable in children. Only TTP and RT can be evaluated as absolute values in seconds. We believe the reason for this is the dependence of the quantitative values on the amount of ultrasound contrast media applied . But theIn our case, we assessed our decision to the operation management by the result of the MRI with contrast enhanced media and DWI. DynCEUS did not play a role in the treatment decision. The target for pediatric surgeons is always to perform an ovary sparing surgery (OSS) according to the modern surgery strategies . HoweverWe think that dynCEUS could be a helpful tool in differentiating unclear lesions on the ovary, including mature and immature teratomas. However, dynCEUS cannot be the only examination modality, it is always only one component in the overall imaging. In addition, a high level of experience in the use of contrast-enhanced ultrasound is required. Rather, dynamic CEUS could be quite helpful to increase the diagnostic confidence for the presence of immature teratoma. For this purpose, we assess the accumulation of the ultrasound contrast media in the ovary over time and the shape of the vascular architecture. Very bizarre vessels seem to be indicative of a more malignant lesion. Thus, in the second case of the girl with an immature teratoma showed a rather chaotic image during the contrast media inflow compared to the first case with a mature teratoma. This image corresponds to underlying vessels formed in malignant lesions by tumor-angioneogenesis. Such tumor vessels, in contrast to non-pathological vessels, have different characteristics that have an impact on perfusion. Our data show that a longer absolute and relative RT exists in the solid part of the immature teratoma compared to the mature teratoma. The contrast microbubbles in the immature teratoma take a longer time to reach maximal accumulation. The reduced flow velocity of contrast microbubbles in pathologic vessels may be due to the aging, corkscrew-like vessel shape, and branching of tumor endothelial cells across the vessel lumen . HoweverThese two cases teratoma demonstrate that dynamic contrast-enhanced ultrasound with quantitative analysis can help differentiate ovarian masses in children and adolescents. Dynamic CEUS meets the requirements for imaging that is easy and quick to perform. It can be a helpful additional tool in the differentiation of tumors. Thus, it can have a complementary contribution in the planning of a ovarian sparing surgery. However, further pediatric studies of quantitative CEUS are needed to characterize typical patterns of findings, receive more data and to improve pre-operative predictive accuracy in the evaluation of ovarian lesions.The original contributions presented in the study are included in the article/supplementary material, further inquiries can be directed to the corresponding author.The studies involving human participants were reviewed and approved by Ethikkommission Universitätsklinikum Jena (2021-2102). Written informed consent to participate in this study was provided by the participants' legal guardian/next of kin.All authors listed have made a substantial, direct and intellectual contribution to the work, and approved it for publication.The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.


We present a case of a 69-year-old man who had localized pleural metastasis without other organ metastases after nephrectomy for right renal cell carcinoma (RCC). He complained of respiratory symptoms for more than two years after the operation and was confirmed to have right pleural effusion and multiple pleural masses on computed tomography (CT). There were no abnormal findings in the other organs, but the pleural mass gradually increased in size on CT. We suspected malignant tumors such as malignant pleural mesothelioma and synovial sarcoma in addition to RCC metastasis. Finally, we performed surgical resection of the pleural mass under general anesthesia, and we diagnosed pathologically as metastasis from RCC. Distant metastases of RCC are common in the lungs, bones, brain, and liver. To our knowledge, localized pleural metastases from RCC is rare. Saito reported that 154 of 1451 12%) autopsy cases of RCC had pleural metastases, but none of them had localized pleural metastasis % autopsy. This pl2We encountered a 69-year-old man who had localized right pleural metastases after undergoing nephrectomy in September 2017 for right RCC (8.0 × 7.0 × 7.0 cm), clinical stage 3, clear cell type histology, grade 2 A–2C. The3According to a previous systematic review, complete surgical resection is expected to improve the survival rate of patients with distant metastases . However3/year, and tumors <4 cm show even lower growth rates [In general, RCC has a mean diameter growth rate of 0.59 cm/year and volume growth rate of 19.1 cmth rates . HoweverWith regard to treatment, IFN and interleukin (IL) have been used for advanced RCC, but their reported response rates have been low at 5%–27% ,9. At pr4Localized metastatic pleural mass secondary to RCC is extremely rare, and most cases have been treated by IFN, IL, and chemotherapy not surgery. More information about the therapeutic effects and prognosis of cases of pleural metastases from RCC needs to be collected.This research received no specific grant from any funding agency in the public commercial, or not-for-profit sectors.Informed consent was obtained from the patient and it is available upon request.Z.S. designed the study and drafted the manuscript. K.H., S.N., K.T., N.T., M.Y., K.K., J.H., M.M., K.K. contributed to review of this manuscript. All authors read and approved the final manuscript.The authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper.


Introduction: Cystic sellar salivary gland-like lesions (CSSLs) are exceedingly rare, with fewer than a dozen case reports. They contain amorphous colloid identical to Rathke cleft cyst contents, but the cyst wall additionally shows cohesive aggregates of benign salivary glands. We report three new examples. Materials and methods: Two cases were seen at University of Colorado Denver and one at Memorial Sloan Kettering (MSK). Molecular testing was attempted on two of three. Results: Case 1 is a 20-year-old female who presented with panhypopituitarism and was found to have a suprasellar mass that proved to be a CSSL. She received no postoperative adjuvant therapy, but recurrence of headaches and blurred vision 2 years later prompted return to medical attention. A much smaller local cyst recurrence was now accompanied by a thickened, bulbous infundibular stalk. Second resection yielded a gliotic infundibular stalk and amorphous mucin, but no residual salivary-like glands. She is without further recurrence on 6-year follow-up. Case 2 is a 29-year-old female with headache; while seen initially at a tertiary care center, diagnosis was only made after consultation at MSK. Case 3 is 68-year-old female who had originally presented with apoplexy to an outside hospital 7 years prior to surgery and diagnosis. Molecular testing was uninformative on case 1 and negative for mutations or fusions on case 3. Conclusion: Few pathologists or neuropathologists have encountered CSSLs in their practices; case 1 produced recurrence and significant infundibular stalk damage, and case 3 originally manifested apoplexy, features not previously reported.  Microscopic tumors of a few, well-formed salivary acini, lined by low cuboidal epithelium, were recognized to occur in the normal pituitary gland by Erdheim in 1940 . These iLarger, symptomatic, mass-like tumors of more numerous, yet still histologically benign, salivary gland-like tissue in the sellar region are far less common, with less than a dozen case reports in the literature , 13, 14.Although symptomatic cystic sellar salivary-gland like lesions (CSSLs) usually contain amorphous colloid identical to Rathke cleft cyst (RCC) contents, they additionally consist of cohesive mass-like tumors of benign salivary glands within the cyst wall. Interestingly, CSSL is only one of several types of salivary gland lesions that can result from these small, misplaced, heterotopic rests. Indeed, a detailed 1997 description of six cases in a paper bearing the name of “Salivary gland-like tumors of the sellar region” actually consisted of diverse tumors – not the entity of CSSLs reported in the current paper . InsteadIn the current study, we detail three new patients with symptomatic CSSLs, two of whom presented with unique clinical features, including one with clinical recurrence and 6-year follow-up after the second surgery. In addition, for the first time, mutational and fusion testing was attempted on two of the three CSSLs in which material was available. The personal files of the senior authors from each institution  were searched for examples of CSSLs. These were the only CSSLs that each author had seen in their 25+ year practices at each site, indicating the rarity of the entity. Both institutions serve as tertiary care centers for neuro-oncology and pituitary disorders. In addition, informal inquiries made to neuropathologists at several other very large academic centers in North America, in an attempt to increase the numbers of cases in this report, proved futile. Namely, even at many places with large numbers of pituitary cases, CSSLs have not been readily observed. The clinical, neuroimaging, and endocrinological information from the two cases at UCD were obtained from the medical record by the clinicians originally involved in the patient’s care . Limited clinical history was supplied by the referring neuropathologist for the MSK case. Genetic testing was attempted on case 1; a tissue block was submitted to Foundation Medicine, Cambridge, MA, USA. On case 3, fusion and mutational testing was conducted at the Colorado Molecular Correlates Laboratory. Mutational analysis was performed using the ArcherDx Variant Plex Solid Tumor kit  with added custom primers. Libraries were sequenced on the Illumina MiSeq using the v3 kit or the Illumina Next Seq using the Mid-Output kit . Raw sequencing data was processed for mutation calls using the ArcherDx Analysis platform v5.1.2. Gene fusion analysis was performed using the ArcherDx Fusion Plex Solid Tumor kit. Libraries were sequenced on the Illumina MiSeq using the v3 kit or the Illumina Next Seq using the Mid-Output kit. Raw sequencing data was processed for fusion calls using the ArcherDx Analysis platform v4.1.1.7. A list of mutations and fusions covered by these two methods is supplied in the online The patient is a 22-year-old female who was initially seen at UCD in 2011 when she was diagnosed with panhypopituitarism, diabetes insipidus, hydrocephalus, and a sellar/suprasellar mass. Magnetic resonance imaging (MRI) showed a large sellar/suprasellar mass measuring up to 2.4 × 2.2 × 2.4 cm, with regional mass effect as described above. There was internal heterogeneity of T2 signal within the mass and thin peripheral enhancement on T1-weighted studies with gadolinium, leading to primary considerations of a papillary type craniopharyngioma or possibly a Rathke’s cleft cyst a, b. TheThere was moderate lateral and marked third ventriculomegaly which appeared to relate to focal stenosis of the cerebral aqueduct a,b. TherHistology was that of CSSL, with ~ 90% of the specimen composed of acellular amorphous eosinophilic cyst contents typical of RCC c, with tExtensive post-operative evaluation by endocrinology demonstrated persistent panhypopituitarism and diabetes insipidus which had predated her surgery. She was treated with DDAVP, prednisone, estrogen/progesterone hormone replacement therapy, and Synthroid. Interval MRI scans from early-2012, mid-2012, and early-2013 showed that the original cystic lesion had resolved. Two years after initial surgery, she developed marked fatigue, excessive daytime somnolence, and increased generalized headaches. She also endorsed subjective blurred vision, but no peripheral vision defects. Neuroimaging studies now showed a considerably smaller cystic lesion than 2 years previously, which was confined to the pituitary stalk. The enhancing lesion along the pituitary stalk measured 7 × 14 × 6 mm; there was no intrasellar cystic component a. The etA right frontal craniotomy with exploration of the pituitary stalk was undertaken. Intraoperatively, the neurosurgeon could visualize the significantly widened pituitary stalk which appeared to have a grayish discoloration. The arachnoid was dissected away from both the right- and left-hand sides of the stalk, and tumor was removed in fragments. The tumor manifested a very firm mucinous component confined solely to the pituitary stalk, which shelled out from the internal aspect of the stalk. As the mucinous material was removed, the stalk collapsed unto itself, into a thin rim of tissue. There were noted to be striations consistent with the residual pituitary stalk, but the residual stalk was markedly thin and very abnormal. Ultimately, excisional biopsy of the stalk was performed and the lesion was felt to be “generously debulked and possibly gross totally resected”. BRAF mutation was detected. Histological examination revealed 100% of the resection material to be amorphous eosinophilic colloid, this time without salivary gland-like tissue. The resection specimen of the infundibular stalk revealed that the bulbous enlargement was due to a central cyst lined by columnar ciliated epithelium (mucinous contents lost to intraoperative suctioning) surrounded by extensive, uniform gliosis on hematoxylin and eosin d. Glial Over the ensuing 6 years, she had not had tumor/cyst recurrence and her panhypopituitarism and diabetes insipidus have been managed by endocrinology. Molecular testing on the block submitted on this case to Foundation Medicine was technically unsuccessful. The patient is a 29-year-old woman with headache and a pituitary mass having solid and cystic components; neuroimaging studies were not available. The neuropathologist who initially encountered the lesion had noted that it was negative for the immunostaining normally expected in a pituitary adenoma, such as chromogranin and synaptophysin. Additional immunostaining for anterior pituitary hormonal markers and transcription factors  was also negative. The case was sent in consultation to MSK where diagnosis of CSSL was made. The excisional biopsies were composed of closely juxtaposed benign salivary-type glands producing mucin a, withouFollow-up has been less than 1 year. The patient is a 68-year-old woman whose diagnosis of symptomatic CSSL was made at UCD. She originally had presented to a physician in another state 7 years previously, with what she described as a fairly acute onset of headache and severe fatigue. She recalled a prolonged work-up that resulted in the discovery of a pituitary mass that was a presumed pituitary macroadenoma with hemorrhage/apoplexy. Full endocrinological work-up had revealed panhypopituitarism and she had been on anterior and posterior hormone replacement since then, including DDAVP (desmopressin). No surgical exploration was undertaken at that time. She subsequently developed worsening headaches, and came to UCD for evaluation and surgery. Panhypopituitarism was confirmed, and MRI revealed a 13.5 × 14.0 × 18.6 mm T1-hyperintense mass that filled and expanded the pituitary fossa and demonstrated significant suprasellar extension, resulting in mass effect on the optic nerves a. SurgerExcisional biopsies histologically showed a large collection of closely juxtaposed, benign salivary-type glands adjacent to a thin fibrotic cyst wall b; no antCSSLs are almost always associated with a RCC component , 10, 11,th example of the entity; this case was also accepted as a CSSL by Stefanits et al. [However, one of the five intrasellar “epithelial cysts” described by Fager and Carter  in 1966,s et al. . Two oths et al.  or Koreas et al. . Howevers et al. . For thes et al. , histopath case of a salivary gland-type collection which was suprasellar and presented with blurred vision and hypothalamic dysfunction was intraoperatively associated with “mucinous fluid within the left optic nerve sheath” [Finally, a related 10 sheath” . This re sheath” . These a sheath” , Kim et  sheath” , and Tat sheath”  also to HUMARA) gene [Absence of mutation in our two cases, however, does not prove that CSSLs are not tumors, i.e., are not neoplastic. Indeed, the most commonly accepted methodology for determining if a growth is monoclonal is testing for X chromosome inactivation and involves determination of whether the same X chromosome has been inactivated within a majority of the cells in a tissue sample (referred to as non-random inactivation and highly suggestive of neoplastic rather than reactive nature of the tissue sample) . X-linkeRA) gene . The volRA) gene , even ifHistologically, the seromucinous glands seen in CSSLs usually do not invoke diagnostic consideration of metastatic tumor, since they are cytologically bland, show perfect preservation of acinar pattern, lack mitotic activity, and are histologically analogous to incidental Erdheim rests – except for the fact that they are several-fold larger and symptom-generating along with the mucin they produce. CSSLs may be embedded within posterior pituitary tissue, as in two of our cases and the case of Kim et al. , or withAs noted previously, symptomatic CSSLs are far rarer than incidental small heterotopic, non-mass producing tumors of a few salivary acini found in autopsy pituitary glands . It is pEctopic/heterotopic salivary gland tissue is not unique to the sellar region. Identical salivary tissue is not infrequently found in extracranial sites, especially in the neck area . The emThe most unique aspect of the current report is the recurrence of symptoms in our case 1 and the apoplectic-like clinical presentation in case 3, neither of which has been previously reported. Case 1 had a recurrence 2 years after her initial transnasal sellar cyst drainage of a much smaller, mucin-filled cyst now confined to the pituitary stalk, and no longer within the sella. While the second surgery, including resection of the pituitary stalk, yielded only the cyst content and not the salivary-type glands, one might speculate that this component had been removed completely at the time of the first surgery and had originally been the major source of the mucinous accumulation that had yielded the original massive 2.4 × 2.2 × 2.4 cm lesion. In at least cases 1 and 3, the size of the mucin-filled cyst was considerable. However, with only a few cases, it would be incorrect to conclude that all CSSLs cysts are larger, or that the mucinous contents produced by the salivary type glands more abundant, than if the lesions had been only pure RCCs. Case 2 is less unique from a clinical standpoint, but nevertheless warrants including due to the rarity of this entity. The second case informs endocrinologists, neuroradiologists, as well as neurosurgeons, pathologists, and neuropathologists involved in the care of patients with sellar region masses that CSSLs do exist, even if they are too rare to enter into differential diagnosis. Indeed, despite the recognition of these rare lesions over 50 years ago, CSSLs are seldom encountered, even by experts. We add these three new cases to the literature and provide a literature review that places them into perspective. The authors thank Ms. Jennifer Platte for manuscript preparation, Ms. Lisa Litzenberger for photographic expertise, and Dr. Mark Ewalt from the Colorado Molecular Correlates Laboratory for mutational/fusion testing on case 3. None. The authors declare no conflict of interest. Supplemental material


Nowadays, videothoracoscopic lobectomy is accepted as the standard treatment method in early-stage lung cancer. Sleeve lobectomy, which is an alternative to pneumonectomy in centrally located tumors or peribronchial tumor infiltration, was previously performed via thoracotomy, but it can be performed by video-assisted thoracoscopic surgery (VATS) in recent years. In this method, without compromising oncological principles, a more comfortable and less morbid postoperative process can be provided to the patient compared to thoracotomy. Here, we aimed the presenting two cases that underwent VATS sleeve lobectomy for non-small cell lung cancer and their results. Sleeve lung resection, a parenchyma-sparing lung resection technique, has more advantages than pneumonectomy in terms of early and late mortality/morbidity . Many stCase 1A 75-year-old male patient was admitted to us with exertional dyspnea and cough complaints. Thoracic CT showed a right upper lobe collapse and a 25 mm diameter mass located in the right upper lobe bronchus Figure . A pathoCase 2A 49-year-old female patient was admitted to us with cough complaints that had been ongoing for eight months. The thoracic CT showed a vegetant mass with a 3 x 2 diameter arising from the upper lobe bronchus and extending into the right main bronchus Figure . HistopaIn this report, we reviewed the current literature because our two cases underwent VATS bronchial sleeve lobectomies. The first VATS sleeve lobectomy reported in the literature was in 2002. In the following years, more complicated surgeries, such as uniportal VATS sleeve lobectomy and bronchovascular VATS sleeve lobectomy, were reported as case series . In VATSVATS sleeve lobectomy can be preferred in indicated cases, such as endobronchial lesions or peribronchial lymph node infiltration. Although the technical details, perioperative results, and short-term survival rates are satisfactory, there is a need for controlled studies with larger case series to evaluate the long-term outcomes of the oncologic cases.


Fibroepithelial polyps or acrochordons are benign skin tumors of mesenchymal and ectodermal origin. They are seen in 25% of the population, and their frequency increases with age. They are often found in areas with skin folds, such as the neck, axilla, submandibular, or inguinal region. However, they can also be located in the genital tract. Hormone imbalances may facilitate the development of fibroepithelial polyp s . Larger lesions are likely to arise from the proliferation of mesenchymal cells within the hormonally sensitive subepithelial stromal layer of the lower. Generally, their size does not exceed 5 millimeters. We present a 28-year-old patient with multiple giant fibroepithelial polyps with size of 20 × 12 × 8 cm and 9 × 4 × 2 cm, located on both sides of her vulva. Herein, we presented our patient along with the review of current literature pertaining to the diagnosis and the treatment of fibroepithelial polyps (FEPs) and the factors leading to excessive growth. Fibroepithelial polyps or acrochordons are benign skin tumors of mesenchymal and ectodermal origin. They occur in 25% of the population, and their frequency increases with age , 2. ThesIn this case report, we present a case of multiple giant fibroepithelial polyps on the vulva.nd child four years prior, from the size of a thumb that gradually increased until its current size. There were complaints of leucorrhoea. There was no history of vaginal bleeding, trauma, sexually transmitted disease, or any medical or surgical history. Her menstrual history was unremarkable. She used hormonal contraception  for three years. She was a nonsmoker and denied alcohol or drug use.A P2A0 28-year-old woman came to the Gynecologic Outpatient Clinic in Hasan Sadikin General Hospital in August 2020 with a chief complaint of a large mass on her left vulva since four years earlier. The patient first noticed an itchy swelling on her left vulva that enlarged after she delivered her 22. On gynecological examination, we found a large, firm, nontender, nonulcerating, skin-colored pedunculated mass, measuring 15 × 10 × 8 cm protruding from the left labium majora  and using hormonal contraception suggested that the two factors might have influenced the tumor growth [It is not known exactly what triggered the growth of the fibroepithelial polyps that are located in other locations and reached huge sizes . Howeverr growth .Although the risk of malignancy and recurrence are very low, malignancy must be excluded by tissue biopsy. Botryoid embryonal rhabdomyosarcoma is the main differential diagnosis, and differentiated sarcomas from a fibroepithelial stromal polyp may be difficult . A distiIdeal treatment for this lesion is complete excision and long-term follow-up to detect recurrence at the earliest. Although fibroepithelial polyps are benign skin tumors, they tend to regrow if not totally excised . They caLarge fibroepithelial polyp of the vulvar region is a rare benign tumor that can be misinterpreted as malignant owing to its wide range of morphological appearances. Expert pathological interpretation may be necessary to exclude atypical tumors and malignant neoplasms or to indicate proper treatment.


Cystic lymphangioma (CL) is a rare benign tumor that mainly occurs in the neck and axilla and usually occurs in children, whereas lymphangiosarcoma (LAS) is a rare invasive tumor, usually secondary to chronic lymphedema caused by various causes, with a rare malignant transformation from CL. We presented the case of a 63-year-old woman who underwent four surgical excisions for multiple recurrence of CL in the right groin. The changes of imaging and pathological examination revealed the unusual process of its gradual malignant transformation into LAS. We followed up the patient for 16 years, and she eventually died of LAS complications. Lymphangioma (LA) is a rare benign tumor. The common clinical type of LA is cystic lymphangioma (CL) , and surA 63-year-old woman was hospitalized for the fourth recurrence of a right inguinal tumor and abdominal distension. The patient underwent the first surgical resection of the right inguinal mass in 2002 and was diagnosed with CL by pathological examination. Subsequently, the patient was hospitalized in 2008 and 2013 because of tumor recurrence. Preoperative imaging examination showed right inguinal polycystic lesions with uniform signal/density, low signal intensity on T1-weighted imaging, high signal intensity on T2-weighted imaging, and slender septum 2. No enIn 2016, the patient was hospitalized for surgical resection because of the third recurrence of the tumor. On the right side of the groin area, the children’s skull size irregular mass was palpable. It extended to the right lower abdomen, with soft texture, unclear boundary, no tenderness, and poor activity, which did not include the abdomen when lying down. Moreover, the patient’s lower limbs were not swollen. Magnetic resonance imaging (MRI) revealed that the right inguinal cystic lesion extended upward to the retroperitoneum with uneven signal intensity and thickened low-signal intensity septum  for the publication of any potentially identifiable images or data included in this article.XX: designed the study. HY and JL: collected and analyzed the data. HY, QM, and LZ: manuscript writing. XX: reviewed and edited the manuscript. All authors contributed to the article and approved the submitted version.This research was funded by Scientific Research Project of Hubei Provincial Health Commission (WJ2021F014) and Wuhan Municipal Health Commission (WX20Z46).The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.All claims expressed in this article are solely those of the authors and do not necessarily represent those of their affiliated organizations, or those of the publisher, the editors and the reviewers. Any product that may be evaluated in this article, or claim that may be made by its manufacturer, is not guaranteed or endorsed by the publisher.


The aim of this article is to describe two spontaneous cases of appendicitis in pet rabbits, to describe therapeutic appendectomy, and to discuss the microbial flora of the inflamed appendix. A 5-month-old intact female and a 16-month-old, neutered male were presented to the veterinary clinic with restlessness, anorexia, and reduced faecal output. The main clinical findings were restlessness, severe discomfort on abdominal palpation, a mid-abdominal palpable tubulous mass and an elevated rectal temperature. Blood analyses showed lymphocytosis, monocytosis, and hyperglycaemia. Radiography was inconclusive. Abdominal ultrasound revealed a presence of a tubular structure with wall thicknesses of 4.2 and 3.7 mm in the two rabbits, respectively. The tubular structure had a rounded, closed end, and a multilayered wall, suggestive of appendicitis. Due to metabolic acidosis and poor prognosis, the first rabbit was euthanized. In the 16-month-old rabbit, appendectomy was performed. Recovery was uneventful, and 4 h after surgery, the rabbit started to become normally active. Postoperative care consisted of fluid therapy, multimodal analgesia, supportive care and prokinetics. Follow-up examinations at 10 days, 1 month, and at 11 months after the surgery did not show any abnormal clinical or laboratory findings. Histopathological examination of appendices from both rabbits showed gangrenous appendicitis. Aerobic cultivation showed the presence of pure culture of Klebsiella variicola sensitive to enrofloxacin, marbofloxacin, tetracycline, cefuroxime, trimethoprim sulphonamide, neomycin, and gentamicin. Restlessness associated with anorexia, abdominal pain, palpable abdominal mass, hyperglycaemia, lymphocytosis, and elevated rectal temperature may be indicative of inflammation within the gastrointestinal tract. Abdominal ultrasound is recommended in rabbits with showing these clinical signs because radiography can be inconclusive. Appendicitis is a life-threatening condition, which should be included into the list of differential diagnoses; for the rabbit, an acute abdomen and gastrointestinal stasis syndrome and must be treated immediately. K. variicola may be associated with appendicitis in rabbits as a causative agent or in association with appendix intraluminal dysmicrobia.Although laboratory rabbits are commonly used as models of appendicitis in man, spontaneous appendicitis was only described  Blood analyses showed leucocytosis, lymphocytosis, monocytosis, hyperglycaemia, and hyperglobulinaemia . Urine aSupportive treatment was started immediately and consisted of intravenous fluid therapy at 15 ml/kg/h  and a 5-ml bolus of 5% glucose, analgesic drug buprenorphine at 0.03 mg/kg i.m. , a prokinetic drug metoclopramide at 1 mg/kg i.m. , an appetite stimulant pro toto at 2 ml , an antiulcer drug famotidine at 1 mg/kg i.m. , a hepatoprotective drug at 0.1 ml/kg/pro toto , and vitamin C at 30 mg/kg s.c. . Meloxicam at 0.8 mg/kg s.c.  was administered 4 h after the patient admission to the clinic, when it was stabilised with intravenous fluid therapy.2  measurement using a pulse oximeter  attached to the hind-limb fingers.Anaesthesia was induced with medetomidine at 0.02 mg/kg i.m.  and ketamine at 3 mg/kg i.m.  followed by propofol at 0.3 ml to effect . The rabbit was intubated using over the endoscope technique, and anaesthesia was maintained with isoflurane at 1.5–3% . Continual intravenous fluid therapy with Ringerfundin was administered throughout the surgical procedure at a rate of 10 ml/kg/h. Perioperative monitoring consisted of observing respiration by close inspection of the thoracic wall, lung and heart auscultation, ECG monitoring, and pOsacculus rotundus and a thickened processus vermiformis caeci (appendix) with miliary yellowish lesions visible in the wall of these structures  and a thickened ructures . All theIn the 4 h following surgery, the rabbit started to recover and did not show extreme pain on gentle abdominal palpation. Intravenous fluid therapy was continued, and analgesics  were given. To support intestinal motility, apart from metoclopramide, oral itopride at 10 mg/kg q12h  and intravenous lidocaine at 100 μg/ml/h  were also administered. The preoperative supportive treatment was continued for 6 days with fluid therapy. As antibiotics, a combination of marbofloxacin at 10 mg/kg i.m. q24h  and metronidazole at 20 mg/kg i.v. q12h  was used. The rabbit was syringe-fed with recovery diet  and Supreme Science Recovery Plus, Supreme Petfoods, Ipswich, UK). It started to eat on its own and produce faecal pellets 2 days after surgery.After 6 days, the patient was sent to home care with oral itopride at 10 mg/kg q12h, oral meloxicam at 0.8 mg/kg q12h, and oral marbofloxacin at 10 mg/kg q24h, for the next 10 days. Follow-up examinations at 10 days, at 1 month, and at 11 months after the surgery did not show any abnormal clinical or laboratory findings.Klebsiella variicola sensitive to enrofloxacin, marbofloxacin, tetracycline, cefuroxime, trimethoprim sulphonamide, neomycin, and gentamicin and resistance to penicillin G, amoxicillin clavulanate, and bacitracin. Bacteroides sp. was isolated in low amounts and was sensitive to enrofloxacin, marbofloxacin, clindamycin, and metronidazole and resistant to tetracycline, trimethoprim sulphonamide, and penicillin G.Samples of the appendix wall and its necrotic contents were sent for histopathological and bacteriological testing. Histopathological examination of the appendix wall revealed the same findings as in the previous case . AerobicAlthough appendicitis in humans can be caused by direct luminal obstruction , the full range of specific causes remains unknown. Recent theories focus on genetic factors, environmental influences, and infections .In rabbits, the vermiform appendix differs from the vermiform appendix in man. It has a larger lumen, so the possibility of spontaneous obstruction is reduced. It also has a thinner muscularis and a different configuration of the mucosa and lymphoid tissue . No aetiEscherichia coli, Klebsiella pneumoniae, Streptococcus spp., Enterococcus spp., and Pseudomonas aeruginosa  and cephalexin (43%) but not against ceftazidime, ceftiofur, tetracycline, enrofloxacin, gentamicin, and trimethoprim–sulfamethoxazole (Antibiotic treatment (a combination marbofloxacin and metronidazole) was in the presented clinical case report aimed to prevent and treat aerobic and anaerobic infection. Antimicrobial sensitivity revealed sensitivity of hoxazole . Authorshoxazole  and may hoxazole . Use of The diagnostic and therapeutic approach to the acute rabbit abdomen should be the same as other emergency cases in rabbits , 28. GI Lymphocytosis in rabbits is associated with inflammatory changes of lymphoid organs, viral disease, lymphoproliferative disorder, and lead poisoning but is also seen with high adrenaline levels, which can be associated with increased activity and restlessness seen in the two presented rabbits .Blood glucose measurements in clinical case 2 showed hyperglycaemia, which can be due to stress and pain. Harcourt-Brown and Harcourt-Brown  found thA diagnosis of appendicitis in humans is based on abdominal palpation, ultrasound examination (86% sensitivity), and computed tomography imaging (92.3% sensitivity) , 35. In Nicolletti et al.  describeThere is only one published clinical case of appendicitis in a pet rabbit , and theAppendicitis in adults and children is a painful disease and is one of the most common conditions that require emergency abdominal surgery . In normSpontaneous appendicitis classification and guidelines either for medical or surgical treatment of choice are not published in rabbits, but the authors recommend vermiform appendix excision because gangrenous appendicitis was diagnosed in both the presented cases. There was a high risk of subsequent GI tract perforation and death of the patient, especially in more advanced cases of appendicitis .An important aspect of the appendicitis treatment is the pre- and postoperative multimodal supportive care. In the presented case, the rabbit after vermiform appendix excision recovered uneventfully. The rabbit survived 11 months to this date with no further GI tract disorders. Sacculitis cannot be treated using surgical excision, so supportive care and antibiotics are advocated.K. variicola may be associated with appendicitis in rabbits as a causative agent or in association with appendix intraluminal dysmicrobia.In conclusion, restlessness or abnormally active behaviour associated with anorexia, abdominal pain, palpable abdominal mass, hyperglycaemia, lymphocytosis, and elevated rectal temperature may be indicative of a condition that causes inflammation and pain within the GI tract, such as appendicitis. Abdominal ultrasound is recommended in rabbits with GI tract disorders, as radiography can be inconclusive. Sacculitis and appendicitis should be included into the list of differential diagnoses for the rabbit acute abdomen and GI stasis syndrome. The original contributions presented in the study are included in the article/supplementary materials, further inquiries can be directed to the corresponding author/s.VJ was a major contributor in manuscript writing, analysing and interpreting the patient data, surgery, and manuscript writing. AP was a major contributor to the manuscript writing and literature review. ID, KH, and JC contributed to the manuscript writing and literature review. MS analysed and interpreted the patient data regarding histopathological diagnose. All authors have read and approved the final manuscript.This study was supported by the grant of the University of Veterinary Sciences Brno 2021ITA15.The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.All claims expressed in this article are solely those of the authors and do not necessarily represent those of their affiliated organizations, or those of the publisher, the editors and the reviewers. Any product that may be evaluated in this article, or claim that may be made by its manufacturer, is not guaranteed or endorsed by the publisher.


The pancreatic rest, aberrant, or heterotopic pancreas is a normal function pancreas found in the submucosal layer of the greater curvature of the gastric antrum and occasionally in the duodenum. Most of the patients are asymptomatic and the finding is usually incidental. We describe the case of a child with abdominal pain and history of recurrent ulcers that necessitated esophagogastroduodenoscopy and further evaluation with endoscopic ultrasound that confirmed a submucosal lesion consistent with a pancreatic rest. Endoscopic submucosal dissection was performed without complication, and complete symptom resolution was achieved after dissection of the pancreatic rest. A pancreatic rest (PR) is heterotopic pancreatic tissue commonly found in the submucosal layer of the greater curvature of the gastric antrum and occasionally in the duodenum . Most ofA 7-year-old Hispanic female with a history of gastroesophageal reflux disease, recurrent ulcer disease, and vomiting presented with a chief complaint of constipation and second opinion of chronic pressure-like periumbilical abdominal pain. Before her visit, her pain was managed with a bland food diet and her ulcer disease was treated with pantoprazole, without improvement on her pain. Family history includes adenomatous polyps and gastric cancer. The parents reported no changes in appetite, vomiting, diarrhea, or weight loss. Physical exam was normal and polyethylene glycol 3350 was prescribed for constipation. She has a normal serum amylase and lipase. Esophagogastroduodenoscopy (EGD) was performed due to persistent symptoms and a 2 cm umbilicated lesion was identified along the posterior wall of the antrum of the stomach . BiopsieEUS revealed a mixed echogenic structure enclosing an anechoic structure consistent with PR , no signExcised tissue was captured with a net retriever tool and sent to pathology, which revealed focal pancreatic heterotopia with acini, ducts, and islet cells in the subepithelium covered with distorted gastric mucosa, and thermal artifactual; there was no sign of dysplasia or inflammation . The patThe PR, known as aberrant pancreas, functions as a normal pancreas . A PR doMost of the patients are asymptomatic . Less coThe increasing availability of EUS has facilitated a more thorough examination of the lesion before intervention. EUS aids in determining the layer of the stomach wall from which the PR arises and helps characterize the lesion . Some ofMost of the patients are asymptomatic and do not necessarily require resection . Howeveren bloc resection of lesions regardless of tumor size [Historically patients with symptomatic PR underwent surgery of the rest by wedge excision and pyloroplasty or by submucosal enucleation through a gastrostomy . Novel emor size . The promor size . Short-tmor size .In conclusion, we demonstrated a symptomatic pediatric patient with a PR successfully managed with ESD. Historically, clinical symptoms warrant surgical management of the PR. Abdominal pain unresponsive to PPI and history of recurrent ulcers in this patient necessitated EGD and further evaluation with EUS, which diagnosed with a submucosal lesion consistent with a PR. ESD was performed without complication, and complete symptom resolution was achieved after dissection of the PR.A pancreatic rest is heterotopic pancreatic tissue commonly found in the submucosal layer of the stomach. Most of the patients are asymptomatic and the gastrointestinal symptom most commonly associated with pancreatic rest is abdominal pain. Patients usually do not necessarily require resection. However, the differential diagnosis for gastric subepithelial lesions includes tumor. Endoscopic mucosal resection is described as an endoscopic treatment with the advantage of being less invasive and at a lower cost, in the other hand; the endoscopic submucosal dissection can be performed regardless of the size of the tumor.


Background: osteoblastoma is a bone-forming tumor accounting for about 1% of all primary bone tumors and 3% of benign bone tumors. The gold-standard treatment is surgical excision; nevertheless, minimally invasive radiological techniques such as thermoablation and, more recently, high intensity focused ultrasound are gaining more importance. The aim of the present paper is to analyze surgical indications based on our experience and on the evidences in the literature. Methods: all patients affected by osteoblastoma who underwent surgical excision in January 2009 and December 2018 were reviewed; eleven patients were enrolled in the study. The epidemiological aspects, size of the disease and site of onset, symptoms, surgery type, indications, and results are reported for every case. Results: all treatments were based on a preoperative diagnosis; pain was constant in all cases. Intralesional surgeries were performed in 9 out of 11 cases; the remaining 2 cases underwent wide resection. No early or late complications occurred after the surgical procedure. The indications for surgery were lesions very close to nerves or joints, unclear diagnosis, risk of fracture, lesion too large for radiofrequency thermoablation, or failure of minimally invasive treatments. At a medium follow-up of 88 months, no local recurrences were verified. Conclusions: osteoblastoma is a rare tumor with difficult diagnosis. Identification is based on symptoms, imaging, and histology. When possible, minimally invasive techniques is preferred for treatment but surgery is still considered the gold standard. Osteoblastoma (OBL) is a bone-forming tumor accounting for about 1% of all primary bone tumors and 3% of benign bone tumors .It usually arises in adolescence and in juvenile ages (second to third decades of life) with a male predominance of 2.5 to 1, although more aggressive forms are described in older patients ,4.It involves the spine in about 32% of cases, usually arising in the posterior elements, but it can also develop from the vertebral body, albeit rarely. The other most common sites are the femur (12%), the jaw (11%), tibia (10%), and the foot and ankle (9%) .A diagnosis in essential for treatment and is based on symptoms, imaging, and histology.Sometimes OBL is asymptomatic and is discovered during exams performed for other reasons; when symptomatic, swelling and localized pain are common, but unlike osteoid osteoma, it is less responsive to NSAIDs. Neurological symptoms can also be present in spine sites. Additionally, systemic symptoms such as fever and weight loss can be present ,7.Upon imaging, OBL can be very similar to osteoid osteoma: a round or oval radiolucent lesion with well-defined margins and variable sclerosis, which usually increases with patient age . SometimHistology reveals the mineralized bone trabeculae of mature osteocytes with normal nuclei and abundant cytoplasm, surrounded by a single layer of osteoblasts. The aspect is very similar to that of osteoid osteoma; nevertheless, more multinucleated giant cells can be present .In the past, it was considered of the same class as osteoid osteoma and its size was the characteristic that distinguishes between the two diseases; indeed, a diameter inferior to 2 cm was considered suggestive of osteoid osteoma, while a superior diameter was indicative of OBL .While some authors still sustain that it belongs to the same family as osteoid osteoma, today, it is more frequently considered an independent nosological entity ,5,9; altIn the past, the treatment of OO was surgical excision; today, minimally invasive radiological techniques, such as thermoablation and, more recently, high intensity focused ultrasound (HiFu), are considered the gold standard ,11,12.For OBL, due to its greater size, surgery still plays an important role, even if thermoablation and Hifu can be considered case by case, especially for small lesions ,14.Indeed, Arrigoni et all, in 2018, published a series of 11 patients affected by OBL of the spine who underwent radiofrequency ablation, obtaining total relief in all cases ; the samThe aim of the present paper is to analyze surgical indications based on our experience and on the evidences in the literature.All patients affected by OBL who underwent surgical excision in a tertiary research hospital in January 2009 and December 2018 were reviewed. Eleven patients, nine males and two females, with an average age of 23 years  were enrolled in the study . All casDiagnoses were performed based on the histologies, reviewed by expert pathologists trained in musculoskeletal tumors, imaging, and symptoms. They were distinguished from OO because, although histologies are similar, OBLs usually present more multinucleated giant cells than OOs; lesions bigger than 2 cm with a no characteristic pain  were considered OBLs.Enneking staging was used to quantify the aggressiveness . It distAll patients were preoperatively studied with X-ray, MRI, and CT scans to better understand the anatomical characteristics of the diseases and to find the best therapeutic approach. In unclear cases, FDG-PET or SPECT were performed.Surgeries were performed by the same surgical team trained in musculoskeletal oncology; antibiotic prophylaxis was performed with 1 g of cephazolin at three administrations per die and prolonged for three days.The postoperative rehabilitative protocol was discussed case by case, considering the patient’s specific characteristics. Postoperative X-rays were performed on all patients. X-rays and gadolinium-enhanced MRIs were repeated at 40 days and every 6 months for the first three years and then every year.The epidemiological aspects, size of the disease and site of onset, symptoms, surgery type, indications, and results are reported for every case.The study was conducted in accordance with the Declaration of Helsinki Ethical Principles and Good Clinical Practices. All patients provided their consent to use of their data for research purposes and publication.The patients’ main characteristics and results are reported in The present series was composed of 11 patients, 9 males and 2 females, with an average age of 23 years  . AccordiDiagnoses were performed using CT-guided trocar biopsy in 8 out of 11 cases, in which the diagnosis was unclear in 3 cases but a benign nature was assessed; in two cases, the diagnosis was intraoperative; and in the last case, the diagnosis was made during a first unsuccessful surgery performed in a non-specialized center.The most frequent symptom was pain, complained by all patients; the patient with OBL of the D3 pedicle also presented irradiated pain (case 7).Intralesional surgeries were performed in 9 out of 11 cases ; in the two remaining cases, wide resection was performed.In 6 out of 11 cases, reconstruction was performed: in 3 cases, with homoplastic bone chips  and, in 2 cases, with autoplastic bone harvested from the iliac crest (cases 8 and 9). Reconstruction was not necessary in the other cases.All patients reported pain resolution at the first postoperative control about 40 days from surgeries.No early or late complications occurred after the surgical procedures.Our pathological findings confirmed the preoperative and intraoperative diagnoses in all cases. The histological examination showed a sharply demarcated lesion composed of irregular interconnecting trabeculae of woven bone rimmed by plump osteoblasts and scattered osteoclasts with intervening loose fibrovascular tissue. Scattered mitoses with no atypical forms were observed.Surgical removal was indicated in three cases because the lesion was very close to their nerves so thermoablation was judged to be too dangerous . In two cases, the diagnosis was unclear so surgery and an intraoperative histological examination were preferred (cases 2 and 4). In one case, there was a risk of fracture (case 3) so surgical reconstruction was necessary. In one case, the lesion was too large for RF (case 5). In one case, the OBL was near the joint (case 8), and in one case, surgery was performed after RF failure (case 9). In the last two cases, OBL was close to the nerves and the diagnosis was unclear so surgery was highly recommended (cases 6 and 10).The medium follow-up was 88 months . At the last follow-up, all patients were alive without evidence of local disease.The gold-standard treatment for OBL is surgery; generally, intralesional surgery is suggested for grade 2 lesions A, whereaRadiotherapy is also considered for the treatment of OBLs located in the spine or sacrum, where wide resection is difficult and the risk of complications and loss of function is consistent ,17.In the last decade, the improvement of minimally invasive radiological techniques has introduced valid alternatives that must be considered, moreover for small lesions that are not at risk of fracture and far from neurovascular bundles. While in OO, imaging is very characteristic and treatment can be performed without histology, OBL presents a less clear aspect and pain is not as typical as in OO. The treatment has to be based on a diagnosis performed on the histological and clinical aspects: lesions with irregular shape, size bigger than two centimeters, pain not nocturnal and not strictly responding to acetylsalicylic acid, and coherence with a diagnosis of OBL.In our series, biopsy was performed in all cases, in which three cases did not have clear diagnoses although a benign nature was demonstrated. Before proceeding in the curettage, the intraoperative biopsy confirmed our hypothesis.In the presented series, a proximity to nerves was the main indication for surgery; indeed, thermoablation can cause them damage. Additionally, tumor size and shape have to be considered: an irregular shape can be a contraindication for minimally invasive treatment, such as a risk of fracture. In case 3, we preferred to surgically remove the tumor and then to fill the cavity with a homograft to decrease the risk of great trochanter avulsion . LocalizIn the following paragraphs, OBLs are discussed based on the site of onset starting from the most frequent sites, spine and long bones.The most frequent site is the vertebral column (28–36%), mainly the posterior elements and pedicles B 18]. O. O18]. OThe surgical indication is also based on the aggressiveness of the diagnosis: intralesional surgery is suggested for stage 2 OBL, whereas en-bloc resection is more suitable for stage 3. Wide resection decreases the risk of local recurrence, which could be difficult to treat ,19.In 2012, Boriani et al. published their experience on 51 cases, clarifying indications and suggesting RT for stage 3 lesions where wide resection is unfeasible or unacceptable for functional sacrifices .A possible problem of spinal OBL is localizing it, moreover, when it is little . InsertiCzigléczki et al., analyzing their experience on 31 cases of OBL of the occipito-cervical junction, also suggested preoperative embolization to decrease the risk of bleeding .If the OBL is big and an instability could occur after its removal, stabilization is suggested.Particular attention has to be reserved for lesions located in the sacrum, where the recurrence rate is higher than in other areas, possibly due to the complex anatomy and the presence of the sacral roots  22]..22].Wide resection in large tumors is indicated to decrease the risk of local recurrence, which could be difficult to treat .When OBLs are located in the limbs, an intralesional approach can be considered the best-choice treatment, although, if the lesion is small, thermoablation has to be carefully assessed. Surgery is necessary in cases with a risk of fracture case 3;  because Although OBL is common in the limbs, few series of surgically-treated OBLs are available. In 2017, Angelini et al. published 13 cases from 1975 to 2012 of OBL located in the elbow , of which 12 were surgically treated . They reIn 2018, Moreira et al. published a case of OBL onset two and a half years after a clavicle fracture . It was When located in the hand and in the foot, surgical removal can be indicated; indeed, minimally invasive techniques could damage neurovascular bundles and decrease bone segment resistance in the fingers, in the metacarpi, and in the metatarsi; in the carpus and in the tarsus, thermoablation could damage the nearest bones and joints. In the case of an aggressive appearance, wide excision is advisable .Several case reports about OBL of the tarsus are present in the literature. Duan et al. published a case of arthroscopic removal of a lesion located in the talar neck ; in 2017The ribs are involved in about 5% of all cases , while tAlthough biopsies were not always performed in the literature, our group strictly sustains them. OBLs are localized in the skull in just 2–4% of cases, more frequently in the frontal bone  . SurgeryOBL is a rare tumor with a difficult diagnosis. Identification is based on symptoms, imaging, and histology. When possible, minimally invasive techniques is preferred for treatment. Surgery is still the first choice in the case of lesions near neurovascular bundles or joints, of those located in areas with a risk of fracture, when reconstruction is necessary, when the diagnosis is unclear, of lesions located at sites difficult to treat in the case of local recurrence such as in the spine or pelvis, and of lesions located in the hand and in the foot.Further studies with more consistent series are necessary to clarify the presented results.


Intracranial hemorrhage (ICH) as a unique image finding, is a rare presentation of glioblastoma (GBM), and can pose a diagnostic challenge. Hypertensive vascular changes are responsible for the majority of the ICH cases, where hemorrhage from brain tumors account only for 5.1% to 7.2% of cases and, the etiology seems to be multifactorial. We present a clinical case of a 70-year-old male who came to the emergency department after a syncopal episode at the workplace, associated with nausea and vomiting. Computed tomography scan and magnetic resonance imaging showed intracranial subdural hematoma, subarachnoidal and interventricular hemorrhage without any underlying lesion. Follow-up imaging in one month showed a new ICH with a thick peripheral mass concerning an underlying neoplasm. The patient underwent tumor resection and immunohistochemical staining confirmed glioblastoma. Despite a multiapproach treatment, including, chemotherapy, radiotherapy, and follow-up surgery, the outcome was poor. GBM is a great mimicker and may initially present with unassuming intracranial hemorrhage with a much more sinister hidden diagnosis. A high index of suspicion on initial imaging based on the patient’s demographics with early tissue diagnosis is crucial in arriving at the correct diagnosis. This case reinforces the importance of close interval follow-up in patients with spontaneous ICH, maintaining a high suspicion for brain tumors. To date, GBM remains a poor prognosis despite combined surgery, chemotherapy, and radiotherapy treatment. Glioblastoma (GBM) (previously known as glioblastoma multiforme) is a primary brain tumor derived from the neoplastic transformation of glial cells. As per World Health Organization (WHO) classification, GBM is a grade IV glioma and accounts for more than 10,000 cases a year with a poor survival rate, up to 15 months. They usually occur after the age of 40 with a peak at 65 to 75 years and present with gradual, worsening focal neurological deficits, and/or seizure ) and canWe present a case of a 70-year-old male with no pertinent past medical history who presented to the ED after a syncopal episode at the workplace, preceded by headaches and vision changes. The patient was brought to the emergency room where he was found to be in hypertensive emergency, complaining of lightheadedness and episodes of vomiting. The physical exam was unremarkable. Initial CT head showed intracranial subdural hematoma, subarachnoidal and interventricular hemorrhage. Further trauma imaging was negative. Further imaging computed tomography angiography (CTA) head and neck showed stable intracranial hemorrhage, without evidence of aneurysm, high-grade stenosis, or occlusion. Magnetic resonance (MR) brain on initial admission showed multiple subacute resolving hemorrhages. A focal 1.5 cm rim-enhancing lesion about the right posterolateral temporal lobe with associated blooming on gradient recall echo (GRE) and T2 prolongation was presumed to be a subacute hemorrhagic contusion in the setting of trauma Figure -C.A month later, while undergoing follow-up CT head, a new temporal acute hemorrhage was noted and the patient was hospitalized to the medical intensive care unit. His main complaint was intermittent weakness of the left upper extremity since discharge, but the physical and neurological examinations were unremarkable.Due to the unclear etiology of ICH, the patient had a repeat MRI. Much to our surprise, this follow-up MRI showed a significant increase in the size of the previously seen right temporal hematoma with heterogeneous thick peripheral mass-like enhancement with areas of hemorrhage which extended to the adjacent dura. This much uglier appearance and rapidly increasing size were now concerning for either recurrent bleed perhaps into an underlying primary or metastatic hemorrhagic neoplasm.The patient underwent right temporal craniotomy with tumor resection. Pathology showed glioblastoma, WHO grade IV, negative for IDH1 R132H mutation by immunohistochemistry, no epidermal growth factor receptor (EGFR) amplification. Additional molecular workup: positive for Telomerase reverse transcriptase promoter mutation, negative for expression of EGFR deletion mutant. No MGMT methylation detected.The postoperative course was uncomplicated, and the patient was discharged home to follow up with Medical and Radiation Oncology. He completed adjuvant chemoradiation therapy with oral temodar and was on monthly pulsed adjuvant temozolomide (TMZ). Nine months after tumor resection and whilst still on therapy, the patient was admitted for a new-onset seizure. Imaging revealed tumor recurrence at the surgical bed in the temporal lobe with an additional lesion in the right occipital lobe Figures , 3D-K. TWe present a teaching case report of a challenging GBM diagnosis that mimicked intracranial hemorrhage at onset in the setting of new-onset syncope and fall. GBM represents the most extreme tumoral transformation of the glial cell. Overt primary ICH is an infrequent presentation and is associated with abrupt onset of symptoms. Intracranial hemorrhage as part of the hemorrhagic stroke represents the second most common subtype of stroke seen in the emergency setting and accounts for 10% to 20% of cases . HyperteCommon radiologic findings are poorly defined intra-axial lesions with mass effect, involving white matter tracts or large tumors with thick, irregular-enhancing rings and a central necrotic core, sometimes with hemorrhagic changes and perilesional vasogenic edema. The term Multiforme was coined to describe the above presentation in 1926 by Percival Bailey and Harvey Cushing . A retrospective study done by Navi et al. in brain cancer patients showed that intracranial hemorrhage manifest by multifocal intracerebral compartment bleeding (87% of cases) was the most common presentation. The mechanism of bleeding can be related to the direct effect of neoplastic cells affecting blood vessels, or malformed vasculature proliferation due to hypoxia, tumor necrosis, but can also be related to tumor coagulopathy. Instead, hypertension is considered to be responsible for only 8% of parenchymal hemorrhages in cancer patients .According to the literature review for malignancy as a cause for spontaneous intracerebral hemorrhage done by Joseph et al. of 2017, out of 1,445 patients diagnosed with ICH from 1978 to 2010, only 33 patients (2.2%) presented with ICH as the initial findings of an undiagnosed tumor [In a retrospective study done by Thaler et al. normal neuroimaging was found in 8 of 102 patients with GBM. These patients presented with seizures, weakness, or cognitive disturbances. Most of these patients underwent repeat neuroimaging within six months, which then showed progressive disease with confirmatory biopsy [The diagnostic imaging of choice for an intracranial neoplastic disease is MRI with and without contrast. Multitudes of advanced imaging sequences such as functional MRI, diffusion-weighted imaging, diffusion tensor imaging, dynamic contrast-enhanced MRI, perfusion imaging, proton magnetic resonance spectroscopy as well as positron-emission tomography play a key role in the diagnostic process. In a recent meta-analysis published in 2017 by van Dijken, sensitivity and specificity can vary from 68% and 77%, respectively, on anatomical MRI to 91% and 95% on magnetic resonance spectroscopy [Surgery is the initial therapeutic approach for GBM and remains a hallmark in the treatment of malignant brain tumors. GBM remains a poor prognosis despite surgical resection and combined chemotherapy and radiotherapy. To date, excluding Tumor-Treating Fields (TTFields), no new agents improve survival when added to standard therapy. MGMT promoter methylation is predictive of response to Temozolomide, its role in the choice of first-line therapy is currently limited to the elderly GMB patients. No standard of care is established in the recurrent setting. Bevacizumab impacts progression-free survival, but its role in overall survival remains less certain. TMZ re-challenge is a treatment option, especially for O6-methylguanine-DNA methyltransferase promoter methylated GBM .When tumor recurs, treatment options include supportive care, reoperation, re-irradiation, systemic therapies, and combined modality therapy . In thisIn the case of our patient GMB re-occurred nine months after the first resection and the second surgery did not improve overall survival, patient was ultimately enrolled in hospice care and passed away shortly thereafter.GBM is a great mimicker. It may initially present with unassuming intracranial hemorrhage with a much more sinister hidden diagnosis. A high index of suspicion on initial imaging based on the patient’s demographics with early tissue diagnosis is crucial in arriving at the correct diagnosis. Close interval follow-up after surgery is a must since it has a very high rate of recurrence. MRI with and without contrast is the basic workhorse, however advanced MRI imaging such as MRI spectroscopy can be particularly helpful in early diagnosis.


Tumors of the ampulla of Vater are uncommon lesions accounting for only 0.5% of the gastrointestinal tumors. A total of three techniques for ampullary tumors (AT) exist: endoscopic papillectomy (EP), surgical transduodenal ampullectomy (TDA), and partial pancreatoduodenectomy (PD). Scarce articles report the outcomes of TDA.Two Arabic men presented to the hospital with epigastric pain and were subsequently diagnosed with AT. The TDA was performed a few days after the diagnosis. The first patient underwent a partial gastrectomy on the eleventh postoperative day. The second patient underwent endoscopic pyloric re-opening on the eleventh postoperative day to relieve gastric obstruction.Both patients started tolerating food and were discharged home. Further randomized controlled studies assessing long-term complications, efficacy, and efficiency of TDA are now warranted. Tumors of the ampulla of Vater are relatively uncommon lesions accounting for only 0.5% of gastrointestinal tumors . Owing tThe AT could be resected with endoscopy or surgery. The endoscopic techniques are endoscopic papillectomy (EP), or "the larger" endoscopic ampullectomy (EA). There are three surgical techniques ranging from the simplest to the most complex: transduodenal ampullectomy (TDA), surgical ampullectomy with complete resection of the common bile duct, and partial pancreatoduodenectomy (PD) (Whipple's procedure) . The preBenign lesions with a low rate of malignancy are preferably treated with endoscopic procedures . HoweverThe transduodenal ampullectomy is performed in benign lesions not amenable to endoscopic resection post sphincterotomy, and large tumors or lesions with endobiliary extension not accessible with endoscopy. Compared to PD, TDA has lower morbidity and mortality but a higher recurrence rate due to limited tissue resection and lymph node dissection . TherefoDespite the increased interest in performing TDA, the clinical nuances and outcomes of this surgery have not been described clearly, and till now, there's no standardized approach for the technique choice. In this article, we report the presentation, complication, and management via TDA in two patients.Case oneA sixty-seven-year-old Arabic man, who is a non-smoker, hypertensive, diabetic, and hyperlipidemic, presented to the hospital with severe epigastric abdominal pain. Physical examination and laboratory tests were unremarkable except for a mild elevation of the pancreatic enzymes. Liver enzymes and CA19-9 levels were normal. An abdominal CT scan revealed intra- and extra-hepatic biliary tree dilation  showed dilated biliary/pancreatic ducts without stones. The endoscopic retrograde cholangiopancreatography (ERCP) combined with a EUS showed mild double duct dilation with a large edematous friable fungating lesion at the level of the ampulla with no duodenal wall invasion, no pancreatic extension, and no lymph nodes detected. Biopsies taken have confirmed the presence of a benign ampulloma. Subsequently, a sphincterotomy with endobiliary stent insertion was performed.A TDA with intraoperative frozen sections was performed. A complete resection of the ampulla of Vater, the sphincter, the distal part of the common bile duct (CBD), Wirsung duct as well as the duodenal wall around the papilla was done and sent to the histopathology section Figure .On frozen section, the ampulla was grossly expanded and widened with polypoid components; no hard or infiltrative areas were found. Surgical margins were unremarkable except that the CBD margin appeared edematous and thickened. The main pathology was the presence of adenomatous glands along with wide areas of low-grade dysplasia in lining epithelium and glands Figure .The lesion was limited to the mucosa with a mildly inflamed submucosa. The CBD was re-trimmed until reaching negative margins, and both the CBD and pancreatic duct were re-implanted in the duodenum.On the fifth day, clear fluid diet was started. Subsequently, the patient reported having persistent vomiting. The upper gastrointestinal series showed huge gastric dilation with slow emptying of contrast at the level of the bulb of the duodenum (D1) and a mild reflux into the common bile duct, as well as gas with gastroparesis and failure of improvement on intravenous prokinetics. As a result, a partial gastrectomy with a Billroth II anastomosis was performed on the 11th post-operative day. The patient was started afterwards on full fluid diet, tolerated well and was discharged 17 days after the initial ampullectomy.Case twoA seventy-eight-year-old Arabic man, who's a non-smoker, hypertensive, and diabetic, presented to the hospital with weight loss, black tea-colored urine, jaundice, and vomiting for the past three months.The CT scan showed biliary dilatations with pancreatic duct dilatation but no mass was found in the pancreatic head. The common hepatic artery was completely thrombosed. The liver received the arterial blood supply from collaterals arising from the gastroduodenal and superior mesenteric arteries. The patient's abdominal vessels were all calcified. The MRCP revealed a dilated CBD (eight millimeters) with no filling defect. On ERCP combined with EUS a peri-ampullary lesion was found with no direct invasion to the duodenal wall, no pancreatic invasion and no endobiliary extension Figure .Biopsies were taken and showed an adenoma with high grade dysplasia.A transduodenal ampullectomy with total removal of the ampullary complex, reconstruction of the biliary and pancreatic duct over the duodenal wall, and stent placement were done. The frozen section confirmed the presence of an adenocarcinoma mostly of intermediate grade and focal high-grade component Figure .The lesion presented as a focal firm mass at the ampulla and measured 1×1×0.5 cm. The tumor infiltrated the mucosa, submucosa, and the superficial muscularis propria but without vascular invasion. The CBD and pancreatic duct margin sample were exempt of malignant cells and the ductal anatomy was re-implanted. A pyloric exclusion was performed with vertical mattress suturing of the distal antrum with thick monofilament sutures and a side to side Billroth II type gastrojejunostomy was performed.Due to the complete calcification of the supraceliac aorta and splenic artery, the liver could not be revascularized after a pancreatoduodenectomy (PD) Whipple's procedure.On the 10th post-operative day, the patient was not tolerating food. The upper gastrointestinal series and the abdominopelvic CT scan showed gastroparesis and distended stomach, respectively Figure .Thus, a gastroscopy was performed on the 11th post-operative day and confirmed a stenotic pylorus. Consequently, endoscopic pyloric re-opening was performed. The patient was discharged on the 18th post-operative day.This article summarizes the findings of two rare cases of ATs; a benign ampulloma and a periampullary malignant lesion.Classically, TDA is geared towards low grade dysplastic ampullary adenomas, whereas EP is performed in periampullary neoplasms sparing the common bile duct and the pancreatic duct , 6. AccoThe delayed gastric emptying reported in case two could be induced by worsened diabetes post-surgery. An inverse relation between glucose concentration and gastric emptying exists, and acute variation in glucose concentration significantly affects gastric emptying . The patThe partial gastrectomy performed in the first patient has hampered the endoscopic follow-up of the patient’s course. Therefore, axial imaging and Ca19-9 levels were used to follow the disease progression.Kim et al. reported the recurrence of an AT post-TDA . The comScarce articles describe the TDA procedure; however, EP is widely expanded in the literature . The EP Surgical transduodenal ampullectomy is rarely performed in our country. The gastroparesis post-ampullectomy reported in the two cases suggests combining gastric drainage to the surgery. None of the patients developed biliary or pancreatic complications. Further randomized control studies assessing long-term complications, efficacy, and efficiency of TDA are now warranted.


The objective is to perform a case report of primary melanoma of urethra in a male patient from a Teaching Hospital in Campinas, SP - Brazil. Male, 79 years of age, with blackened lesion on the meatus of the urethra with 1.5 cm of dimension, that could be palpated on the fossa navicularis region associated to an irregular lesion on the region of and the foreskin. The biopsy showed a malignant neoplasia. A partial penectomy with oncological margin of 2 cm was performed with a bilateral deep inguinal linfadenectomy. A follow up showed no recurrence of the disease. Melanoma is an aggressive tumor with high rates of recurrence and progression. They are malignant tumors arising from melanocyte pigmented cells, that can be mostly cutaneous, but can also occur in various extracutaneous sites where pigment cells are present, such as mucosas, albeit rare.The rarity of the disease often represents a limit to the participation of patients in clinical trials. In fact, studies with immune checkpoint inhibitors, approved in recent years for melanoma treatment, are still underway for mucosal melanoma and. So far, results, despite favorable, have been based on small retrospective series.A 79 yo white male, married, with baseline hypertension, presented for urological consultation due to an altered urinary stream and a visible dark penile lesion on the glans. He noted it 6 moths prior and reported progressive growth. At physical examination, there was a vegetant blackened lesion on the meatus of the urethra, with 1.5 cm, that could be palpated on the fossa navicularis, associated with an irregular lesion on the foreskin . The uriAn incisional biopsy was performed, revealing a proliferation of undifferentiated cells organized in solid or insular patter, with predominant spindle morphology. Nuclei were atypical, with conspicuous nucleoli, and frequent mitotic figures , and an eosinophilic cytoplasm, sometimes with scant brown pigment. As findings were suspicious for melanoma, an immunohistochemical study was performed, which showed positive expression of melanocytic markers .Subsequent clinical staging included thoracic and abdominal tomography with no related findings . A penile MRI reported no evidence of corpus cavernosum or spongiosum invasion. A partial penectomy with bilateral deep inguinal lymphadenectomy  was indicated, with 2.0 cm margin. The surgery proceeded without complications and the patient kept a residual penile shaft of 5.0 cm .Fig. 2PoSurgical pathology  report dPost operatory evolution was satisfactory and patient has no signs of relapsed disease or progression at 24 months of follow up.A large review of literature was performed (1962–2017), and a total of 54 studies reporting penile melanomas. The majority of the authors describe the urethral melanoma as an aggressive disease, with high rates of relapse and progression, with poor prognosis even at earlier stages. In our case report, there was an alteration on the urinary stream and eventual urinary retention, associated to urethral bleeding.The diagnosis was based on the histologic study complemented with immunohistochemistry, performed on an incisional biopsy of the lesion. Urethrocystoscopy may be necessary in cases where an urethral mass can be palpated. Treatment, however, is not a consensus. Some authors report radical penile surgery with extensive lymphadenectomy and even eventual cystoprostatectomy, while others have regarded partial penectomy with oncological margins and inguinal lymphadenectomy as sufficient intervention. More recent studies tend to suggest less aggressive procedures when possible, whereas radical surgeries are reserved for more advanced cases. Concerns about the preservation of anatomical structures and sexual function represent a limitation to the wide surgical margins.uncommon sites include cutaneous and mucosal lesions related to an unusual localization, in specific ethnic groups, in which exposure to UV radiation probably has no pathogenic role. The umbrella term “sun-protected melanomas” refers to mucosal, acral and genital melanomas, but it should be avoided, since these lesions are a heterogeneous pool regarding their genetic profiles and prognosis. The tumor mutational burden of mucosal melanomas is overall lower than that of cutaneous sun-exposed melanomas, which may impact in immunotherapy response rates. Also, a distinct pattern of chromosomal aberrations and a higher rate of copy number alterations have been reported in mucosal melanomas, as well as frequent KIT mutations in urogenital melanomas.Melanomas of Because of its infrequency, diagnosis and treatment of this illness are still a challenge. Primary melanoma of urethra is a rare and aggressive disease. Despite controversies about the extension of surgical intervention, early diagnosis and treatment are fundamental for a successful clinical evolution and patient survival.Not applicable.The authors declare that there are no conflicts of interest.


Haemolymphangioma arising from the small bowel and its mesentery is extremely rare in the clinical setting. To date, only 8 cases of small bowel haemolymphangioma have been reported, and there have been no previously reported cases of haemolymphangioma in the small bowel mesentery (PubMed). The formation of this tumour is mostly congenital, but the exact mechanism is still unclear. As a benign tumour, the presentation of the disease may vary from a simple well-defined cystic lesion to an aggressive ill-defined lesion mimicking malignancy. However, there are no typical symptoms, and preoperative diagnosis is difficult.We present two cases of haemolymphangioma in the small bowel mesentery in a 54-year-old man and a 52-year-old woman. Both of them came to the hospital due to an abdominal mass. In the first case, a cystic teratoma in the left abdominal area was considered after abdominal plain computed tomography (CT) and magnetic resonance imaging (MRI) scans. After taking an enhanced CT scan, a lipoma was considered based on the images. In the second case, cystic masses of the left upper and middle abdomen were observed on abdominal ultrasonography. An abdominal plain CT scan showed an irregular low-density mass in the left upper and middle abdomen. With an enhanced CT scan, haemolymphangioma was considered based on the images. After complete surgical removal, the masses were found to originate from the small bowel mesentery and had not invaded into the peripheral lymphatic tissue. In case 1 in this study, the routine pathology diagnosis was lymphangioma, while in case 2, the diagnosis was haemangioma. The final diagnosis was confirmed to be haemolymphangioma by immunohistochemistry in both cases. No recurrence was evident during 4 months of follow-up. We review the previous case reports of haemolymphangioma in the abdominal cavity and discuss their clinical features, diagnosis, treatment and prognosis.The clinical manifestations of abdominal haemolymphangiomas can vary for both location and size. Abdominal CT examination has important clinical value for haemolymphangioma in the abdominal cavity. The final diagnosis of haemolymphangioma depends on a postoperative pathological examination. In addition, postoperative regular follow-up is necessary. Haemolymphangioma  is a rare type of lymphangioma that shows a mixture of blood vessels and lymphatics. Haemolymphangioma most commonly presents in infants and young children on the body surface. The incidence of haemolymphangioma in adults is low, and there are only a few reports in the literature. In adults, most haemolymphangiomas occur in the head and neck, while only a few cases have been reported to occur in the digestive system, such as the liver , pancreaA 54-year-old Chinese man came to the hospital due to an abdominal mass he had noticed 4 days prior. Abdominal plain CT and MRI scans showed an abdominal cystic mass that was considered to be a cystic teratoma. The patient had a history of inguinal hernia surgery. An abdominal examination showed no positive signs. Routine blood, blood biochemistry, tumour biomarkers , other blood tests and urine analysis were normal. Abdominal enhanced CT revealed , CD34 (blood vessel +), and D2-40 (lymphatic vessel +) positive reactions  and D2-40 (lymphatic vessel +) positive reactions Fig. a–c. The Lymphangiomas are a heterogeneous group of vascular malformations that are comprised of dilated lymphatic spaces. They are benign malformations and are classified into four categories : capillaHaemolymphangioma arising from the small bowel and its mesentery is extremely rare in the clinic. We reviewed the literature by searching for “haemolymphangioma [All Fields] OR haemangiolymphangioma [All Fields]” in PubMed. There have been only 8 cases reported on small bowel haemolymphangioma Table  4–11] a–11 a4–11in this study had undergone laparoscopic total hysterectomy and bilateral salpingectomy three years prior. Surgical injury to her blood vessels and lymphatics has not been ruled out, and it is also possible that the previous surgical exploration missed the haemolymphangioma. In Case The clinical manifestations of abdominal haemolymphangiomas can vary in both location and size. Abdominal pain and an abdominal mass are common symptoms in patients with haemolymphangioma of the pancreas , 3. HaemDue to the difficulty in distinguishing haemolymphangioma from other small bowel diseases, a preoperative diagnosis is extremely hard. Routine blood tests and tumour biomarkers  usually show normal results. Gastrointestinal haemolymphangioma is clinically diagnosed mainly based on endoscopy, such as tumours located in the duodenum , 5, smalin our study underwent a contrast-enhanced CT scan of the abdomen and showed no significant enhancement due to the low proportion of blood vessels in the tumour. In Case The final diagnosis of haemolymphangioma depends on postoperative pathological examination. The architecture of the constituent vessels on routine haematoxylin-eosin (H&E) histological preparations is usually distinguished. Further identification can be made by immunohistochemistry in such cases. Both vascular endothelial cells and lymphatic endothelial cells are CD31- and CD34- positive, while D2-40 is only expressed in lymphangioma and some malignant vascular tumours. Therefore, immunoreactivity for podoplanin with the D2-40 antibody helps to differentiate it from lymphatic endothelial cells . CombineAbdominal haemolymphangiomas are benign lesions, but the tumour growth may be rapid. It can compress the intestinal canal and surrounding parenchymal organs to cause abdominal pain, abdominal distension, bowel obstruction and other acute abdominal symptoms, and the tumour itself may bleed, rupture, or infect. Therefore, once the above signs occur, the patients should be treated in a timely manner. The standard management of haemolymphangioma is surgical resection. Other nonsurgical treatments, including cryotherapy, laser therapy, radiotherapy and local injection of sclerotic agents, compared with surgical treatment, do not show superiority . It has Two cases of haemolymphangioma of the small bowel mesentery were reported here. The clinical features, diagnosis, treatment and prognosis of haemolymphangioma were reviewed in the literature. However, haemolymphangioma of the small bowel mesentery is still poorly understood because it rarely occurs in adults. Clinicians need to improve their awareness of haemolymphangioma.