Myopericytoma of the parotid gland is a rare condition of which preoperative definitive diagnosis is relatively challenging. The purpose of this case report is to highlight the radiologic features of myopericytoma of parotid gland.A 62-year-old man presented with a history of a walnut-size mass in left parotid gland when yawned for half-month, and a 48-year-old woman complaint about a grape-size, painless mass behind the right ear for a month.Radiological examinations suggested that both lesions were cyst-solid mixed lesions with relatively smoothed margins, with or without significant enhancement while the lesion without enhancement had a hemorrhage. Then a diagnosis of benign tumor arising from the parotid gland was made. Final diagnosis of myopericytoma was confirmed by histopathological and immunohistochemical examinations after surgical resection.Both patients underwent excision of the tumor and the superficial parotidectomy with facial nerve preservation.Both patients recovered without any intraoperative or postoperative complication and had no signs of recurrence during a 17-month and 5-year follow-up.Parotid gland myopericytoma is an exceedingly rare tumor which diagnosis can be challenging, and this is the first published report specifying the magnetic resonance features of the disease. The concept of perivascular myoid differentiation was established by Granter. In 2002, the World Health Organization officialized the term “myopericytoma” for use in clinical diagnoses. Myopericytoma arises most commonly in middle adulthood, but may also occur in child. Myopericytoma can be multifocal involving single or multiple anatomic regions, and tends to occur predominantly in the skin and superficial soft tissue of the distal extremities , followed by the head and neck region, and the trunk. Myopericytoma of the parotid gland is very rare. To date, only 5 published cases of myopericytoma arising from the parotid gland have been reported worldwide, and only 1 report described the computed tomography (CT) imaging features in detail. Current report provides a comprehensive description of parotid gland myopericytoma, including its complete clinical course and imaging findings. Reviews of past literature on this rare condition were also discussed.The term myopericytoma was first proposed by Requena et al as an alternative designation for solitary myofibroma derived from myopericytes.1.1The retrospective case report was approved by both patients, as well as the ethics committee of The Second Affiliated Hospital, Zhejiang University School of Medicine.22.1A 62-year-old man was admitted to our hospital following the discovery of a walnut-size mass in left parotid gland when yawned for half a month without any clinical symptoms. His medical history was otherwise unremarkable. Physical examination revealed a soft, painless, well circumscribed, and mobile mass in the left parotid region. Facial nerve function of the patient was normal on both sides. Ultrasound revealed an oval-shape, cyst-solid mixed, 3.0 cm × 2.0 cm mass located in the left parotid gland. Magnetic resonance imaging (MRI) showed a cyst-solid mixed, clearly defined mass in the left parotid gland. The solid component exhibits slightly high signal both on T1-weighted images and T2 weighted images, indicating the possibility of hemorrhage. The area of cystoid variation showed low signal with peripheral high signal on T1-weighted images and high signal on T2-weighted images , often benign with absence of mitoses, pleomorphism, or necrosis. It is believed that the myopericytoma cell of origin is either the pericyte or myofibroblast, both of which exhibit properties of modified smooth muscle cells rather than endothelial cells. However, the maximum diameters of all 7 lesions in the 5 published studies and present report of myopericytoma arising from the parotid gland were 2.5 cm, 5.6 cm, 1.4 cm, 1.2 cm, 0.9 cm, 3.0 cm, and 2.2 cm respectively,,11–14 which is different from the previous research. The lesion with maximum diameters of 5.6 cm is the recurrent focus not long after the last operation and another lesion with maximum diameters of 3.0 cm is with hemorrhage and necrosis. We speculates that recurrence, hemorrhage, and necrosis might be the cause of the enlarged tumor lesion.We reviewed all the previously published cases of parotid gland myopericytoma Table . Previou,11,12 which was similar to the appearance of case two in present report. MR findings were reported by only 1 case with myopericytoma in the parotid gland. Kuczkowski et al reported a case of myopericytoma in the parotid gland showed 2 foci in the left parotid gland with significant heterogeneous enhancement. Myopericytoma arising from other regions also most often appear as an enhanced T1 hypointense, T2 hyperintense mass. The second case in present report had a significant enhancement with mottled non-enhancement area as well. We speculated that the obvious enhancement with or without hemorrhage and necrosis may be the imaging feature for myopericytoma of parotid gland. However, the first case in present report was not enhanced, which is quite different from previous findings. We suppose that this may be related to the hemorrhage in the lesion. To the best of our knowledge, the present report is the first to report hemorrhage in parotid gland myopericytoma. Thus, when a parotid gland mass with a well-defined cyst-solid imaging features and hemorrhage was detected, the diagnosis of myopericytoma should be considered in the differential diagnosis. However, future studies are still needed to verify this guess.Three CT examinations of myopericytoma involving parotid gland showed strong peripheral enhancement and irregular central non-enhanced region after contrast medium administration,The common MRI features of these 2 cases were cyst-solid mixed appearance, and relatively smoothed margins with hyperintense on T2-weighted image in the solid component. We are the first to provide a comprehensive radiological description of parotid gland myopericytoma, including CT and MRI findings. The characteristics of pleomorphic adenoma are hyperintense areas on T2-weighted images with marked enhancement, fibrous capsules, and a lobulated contour. Cystic change and hemorrhage are often seen in larger tumors (>3 cm). Basal cell adenoma shows relatively low signal intensity on T1-weighted images and hypointense to slightly hyperintense on T2-weighted images, with rapid and prolonged enhancement on dynamic scans. Basal cell adenoma sometimes has cystic or hemorrhagic components. Mucoepidermoid carcinoma may be predominantly cystic or mixed cystic with solid mural components. The MRI findings have a tendency to be related to the histological grade. There are some general features suggesting malignancy, such as irregular margins, extra-glandular infiltration, perineural spread, and secondary lymphadenopathies.The differential diagnosis should include tumors with similar MR findings that are commonly seen in the parotid gland. Warthin's tumors usually demonstrated a rounded or lobulated lesion with intermediate signal on both T1-weighted images and T2-weighted images, and focal T2-hyperintense area corresponding to the cystic components. However, the tumor usually shows rapid contrast enhancement and washout on post-contrast T1-weighted images.–4 It is generally accepted that parotid gland myopericytoma should be treated with wide surgical excision to prevent local recurrence. The superficial parotidectomy with facial nerve preservation is known as a widely acceptable procedure. Meanwhile, previous study suggests that via extracapsular dissection may provide improved functional and facial aesthetic outcomes for parotid neoplasms compared with superficial parotidectomy. Radiation has been used in selective cases to decrease the likelihood of recurrence, especially if there has been incomplete tumor excision. Ongoing molecular studies may offer more suitable options for patients with malignant myopericytoma in the future.Myopericytoma usually follows a benign clinical course, in our case, after 17-month and 5-year follow-up, both cases had no signs of recurrence, but local recurrence and malignant myopericytoma have also been reported.4To the best of our knowledge, this is the first report to specify the MRI features of myopericytoma involving the parotid gland. Myopericytoma should be considered during the differential diagnosis of painless, cyst-solid mixed lesions with relatively smoothed margins, high signal intensity on T2-weighted images, and obvious enhancement tumor involving parotid gland. It is worth noting that the tumor may present unenhanced mass due to hemorrhage sometimes.We thank Xiao-Pei Xu for her excellent help in the preparation of the manuscript.Conceptualization: Yao Pan, Lu Chen, Ri-Sheng Yu.Data curation: Yao Pan, Lu Chen, Dan Shi.Formal analysis: Dan Shi, Ying Chen.Methodology: Yao Pan, Lu Chen, Ri-Sheng Yu.Resources: Yao Pan, Lu Chen, Dan Shi, Ying Chen, Ri-Sheng Yu.Supervision: Ying Chen, Ri-Sheng Yu.Writing – original draft: Yao Pan, Lu Chen.Writing – review & editing: Ying Chen, Ri-Sheng Yu.


Background: Epithelioid trophoblastic tumor (ETT) is a rare and newly defined disease, which most commonly occurs in women of reproductive age and can be a sequela of any gestational event. ETT can be present in both intrauterine and extrauterine sites. Case report: A woman of reproductive age, without specific comorbidities and with a single pregnancy and natural childbirth eight years ago, was diagnosed initially with poorly differentiated pleomorphic leiomyosarcoma on the hemostatic uterine curettage. Conclusion: Our case highlights that ETT presents a diagnostic challenge due to its rarity and histologic resemblance to other pathologies. Misdiagnosis delays effective treatment and affects survival. To date, only 8 cases of ETT of the uterus without previous gestational event and normal human chorionic gonadotropin (β-HCG) levels in a 60-year literature survey have been reported. The ETT is a very rare form of gestational trophoblastic neoplasms (GTNs) that grows slowly, produces low levels of beta-subunit of human chorionic gonadotropin (β-hCG), presents late-onset metastatic capability and is resistant to different chemotherapy regimens . In clinA 44-year-old woman , without specific comorbidities, was referred to the Emergency Department at a primary care facility for sudden onset vaginal bleeding, associated with pain of moderate intensity in the lower part of the abdomen. Her last menstrual period occurred two weeks before her presentation, and previous menstrual cycles were regular. She did not smoke or drink alcohol. The patient was otherwise fit and well, with no other significant medical history. She had no significant family history and she is married, with a 12-year-old daughter. The delivery was vaginal, uncomplicated, term pregnancy, without any known difficulties. Also, the patient denied intermenstrual bleeding, post-coital bleeding and recent changes in menstrual cycle.The general condition of the patient appeared to be good. Bimanual examination revealed an anteverted uterus increased in size, firm consistency, painless. There were no masses palpated in the vaginal canal or adnexa. Abdominal-pelvic ultrasound showed an endometrial thickness of 8 mm and multiple hypoechoic intramural masses of different sizes. As the cause of bleeding could not be determined from any coagulation disorder or a scar site, a diagnostic and hemostatic uterine curettage was performed and histology showed poorly differentiated pleomorphic leiomyosarcoma.In April 2020, the patient was submitted to our unit for treatment. After discussion at the oncology multidisciplinary team meeting (MTD), a second pathological opinion was requested.Preoperative investigations and surgical treatmentMicroscopic re-evaluation of slides in the biopsy curettage product was completed with the immunohistochemical examination that established the diagnosis of ETT.Pelvic magnetic resonance imaging (MRI) with gadolinium showed the presence of a large uterus measuring 51  × 79 (max anteroposterior) × 91 mm (transverse), with inhomogeneous structure due to a fundic tumor mass measuring 23 × 32 mm with moderate hypo signal T1, with 2/3 myometrial invasion. The right ovary had multiple peripheral cysts measuring 17 × 12 mm .For the systemic evaluation of the patient, a thoraco-abdominal CT-scan was performed which showed the absence of lesions suspected of secondary determinations.The images and tumor results had been reviewed at the oncology MTD, and the recommendation was surgical treatment. 3, neutrophils—8160 mm3) and normal β-HCG (<2 ng/mL), α-fetoprotein (AFP) and CA125 levels (18.4 UI/mL).Laboratory tests showed mild neutrophilic leukocytosis  was performed . The proAnatomopathological evaluation of the surgical specimenmacroscopic evaluation showed the uterus with irregular contour. The uterine cavity is 4 cm long. At the level of the uterine fundus, with extension to the left horn, there were noted a tumor mass of 3.7/2.5/2 cm, well delimited, pearly white, fasciculate appearance and friable consistency. The tumor protrudes in the uterine serosa and infiltrates over 50% of the thickness of the myometrium. In the right hemibody of the uterus, intramural, there were found multiple nodular tumors, the largest being 2.3 cm in diameter, well defined, pearly white, fasciculate appearance and elastic consistency. The right ovary measured 4.2 cm × 5.1 cm × 1.7 cm with unilocular sero-cytrin ovarian cyst measuring 2.2 cm. The left ovary measured 2.9 cm × 2.2 cm × 0.8 cm with two unilocular sero-cytrin ovarian cysts measuring 0.8 cm.The microscopic evaluation of the surgical specimen showed a uterine nodular tumor with dimensions of 3.5 cm × 2.5 cm × 2 cm developed in the uterine fundus area with extension to the left horn, apparently well delimited both macroscopically and microscopically—pushing type margin.The Intermediate trophoblast tumor cells are arranged in a fibrous eosinophilic material with vesicular nuclei. Tumor cells surround and sometimes completely replace the vascular walls in some medium-sized vascular spaces. Numerous atypical mitoses are identified—19 mitoses/10/high power field. At the level of the uterine serosa, near the tumor formation, an area of its rupture is identified with the exteriorization on the serosa of a small island of tumor cells accompanied by necrosis. The specimen margins were, notably, free of tumor . All theImmunohistochemistry staining of the tumor cells revealed SMA was negative in tumor cells, with positive internal control, desmin was negative in tumor cells with positive internal control, CKiT negative in tumor cells and positive in mast cells, CD 10 intense and diffuse positive in tumor cells and p63 weakly positive, zonal extended in tumor cells .Sixteen months postoperatively, the patient remains disease free according to the paraclinical investigations. She is being monitored monthly with serum β-HCG.Shih and Kurman first described this pathology in 1998 and named it ETT, distinct from placental site trophoblastic tumor (PSTT), which is believed to arise exclusively from chorionic-type intermediate trophoblast . The WorThe incidence of ETT after a term pregnancy is 1:150,000 pregnancies, with evidence of ethnic variation, representing 1–2% of GTN cases ,5.In most of the patients, as in our patient, ETT usually occurs in women of reproductive age, after a full-term delivery. Rarely, it can occur in postmenopausal women. The interval between the ETT and the antecedent pregnancy ranges from several months to many years. Like PSTTs, the antecedent pregnancy like in our case is more often female. The gestational history of our case was represented by full-term delivery. Serum β-HCG levels were almost always raised at the time of diagnosis, Ref.  which isTo date, only 8 cases of ETT of the uterus with normal serum β-hCG in a 61-year literature survey PubMed 1961–2021) have been reported 61–2021 h,11,12,13On macroscopic examination, ETT almost always presents itself as a discrete solitary nodule of up to 5 cm, with well-circumscribed border, with solid, tan to brown surface, often with areas of hemorrhage and necrosis located in the fundus (as in our patient), lower uterine segment, endocervix or lung. Rare cases have included pulmonary  and abdoThe correct diagnosis can only be made following histological examination and confirmed by immunohistochemistry. The most important and difficult part is to distinguish ETTs from PSTT—which may be challenging due to non-specific clinical and biological features, which frequently suggest an ectopic pregnancy. Useful clinical features in the distinguishing diagnoses PSTT and ETT include: vaginal bleeding in ETTs in comparison with abortion and amenorrhea in cases of PSTT.The primary diagnosis of our patient was poorly differentiated pleomorphic leiomyosarcoma. In our case, we took the sequential steps in differentiating diagnoses as presented previously. Central histology review by pathologists with GTN’s expertise is crucial to minimize misdiagnosis, because it may be difficult to differentiate PSTT and ETT from each other, and other types of GTN’s.Microscopically, the tissue from PSTTs have a more infiltrative myoinvasive pattern, an increased sheet-like growth (versus nests and cords), larger cells with more nuclear pleomorphism and multinucleation, prominent and distinctive vascular invasion, strong diffuse staining for hPL and Mel-CAM (CD146), but negative staining for p63 and p40. Extensive or ‘geographic’ necrosis is often present in ETT. Moreover, PSTT is p63 and p40 negative. In consideration to PSTT, indicated above, ETT has shown staining of PD-L1. These results indicate that PD-L1 positive ETTs may gain a benefit from immune checkpoint inhibitor therapy ,18.Immunohistochemical analysis for our case is summarized in However, reviewing the available literature, a few risk factors generally appear to be associated with favorable or unfavorable outcomes. Time from antecedent pregnancy > 4 years, a high mitotic rate > 6/10 HPF, atypia, vascular invasion, myometrial invasion beyond the inner one-third, stage III or IV, diffuse uterine multifocal disease have been associated with worse outcomes for women with ETT ,21. The Overall, this case is a stage I disease (p T1N0) with some good prognostic factors, but at least two poor prognosis factors , which make the prognosis not completely clear (prognostic score = 8). Due to this features, surgery is the most suitable treatment, usually total hysterectomy with or without bilateral salpingo-oophorectomy .A series of recent studies ,25,26 haThe present case underlines the difficulties experienced in diagnosing and treating ETT and highlights the importance of reporting any new cases to add to the understanding of this unusual disease and to achieve a more stable approach for managing these patients.The epithelioid trophoblastic tumors are rare and aggressive malignant uterus tumors, with particular diagnostic challenges, especially when they occur at a long distance from the pregnancy. The immunohistochemical examination of the uterine curettage product may establish the preoperative diagnosis of ETT. It is essential that the clinician be familiar with the lesion, and be anticipative to the possible presence of this disease in order to make early diagnosis.The authors’ opinion is that ETT would be best treated in a tertiary center by an experienced multidisciplinary team.


Background/Aim: Lipomatous meningioma is a rare type of meningioma that is formed as the result of an accumulation of lipids inside the cell due to metabolic activity dysregulation. It differs from other types of meningiomas in its radiological and immunohistochemical characteristics. We report a rare case of a patient treated in our department for this particular type of meningioma who developed a type of migraine with the aura component as the first clinical symptom. Case Report: A 55-year-old woman presented with a migraine and reported having phosphenes in recent years. Head Computed Tomography (CT) and Magnetic Resonance Imaging (MRI) scans were performed; these showed an extensive hypodense and hypointense formation located in the left parieto-occipital region. This formation was implanted in the tentorium region, with a prevailingly adipose-type signal intensity. The patient underwent an occipital craniotomy with the total removal of the lesion. The histological examination indicated a lipomatous metaplastic meningioma. Conclusion: We reported the first case of a lipomatous meningioma presenting with a migraine with a visual aura. Seizures and headaches can be included as possible symptoms. According to the current literature, lipomatous meningiomas affect women more commonly than men. The patient of our reported case presented visual disturbances in the form of a visual aura, which occurred 10 years before finding the meningioma, and surgery dramatically improved the symptoms and quality of life. Meningiomas constitute approximately 36.4% of all CNS neoplasms; these are benign lesions that more frequently involve the female sex. They are classified according to their histological characteristics into different subtypes; lipomatous meningiomas fall within the subtype of metaplastic meningiomas due to the accumulation of adipose tissue within the cells. They have radiological and immunohistochemical characteristics that allow for a specific diagnosis.Headaches and epileptic seizures are the predominant clinical manifestations; this case report presents the first case of a lipomatous meningioma presenting with a migraine with a visual aura.A 55-year-old woman arrived at the emergency department presenting with a closed-head traumatic brain injury due to a car accident. She had a history of migraines with auras consisting of fortification spectra (expanding zigzag pattern) and transient flashing white lights, mostly recurring in the left visual field, followed by a moderate to severe left-sided throbbing headache several minutes later. However, the headache was not predominantly present . The aura episodes lasted several minutes and were associated with nausea, vomiting, photophobia, and headache, which persisted for several hours. These symptoms were relieved by sleeping and administration of oral analgesics. Analysing her medical history, the patient presented with a MIDAS score of 10 points, with grade 2, which corresponds to medium disability. During the three months before the incident, the patient experienced difficulties in carrying out daily activities and she had to take a break from work due to severe episodes of headaches. These episodes were accompanied by the visual aura that caused the patient to stay in bed and to avoid noise and light exposure. Her physical, neurological and fundoscopic examination, including blood pressure and meningeal signs, were normal. Electroencephalography did not reveal any abnormalities. As part of the workup, a CT scan of the brain was obtained. Imaging showed a hypodense area of 40 × 44 mm in size within the left parieto-occipital region. Further evaluation using contrast-enhanced MRI  revealedThe signal intensity of the mass showed characteristics of the adipose-tissue type with a small substantial peripheral component. MRI showed the lesion’s hyperintensity on both T1- and T2-weighted sequences, along with the contrast enhancement after an intravenous Gadolinium injection.The patient underwent a left parieto-occipital craniotomy for a Simpson grade one tumour performing an “en bloc” resection. The lesion, implanted on the superior aspect of the tentorium, appeared well-circumscribed. A clear cleavage plane between the tumour and the normal parenchyma was identified. No brain invasion was found. From intraoperative photography , the tumThe histological examination showed a mixture of meningothelial lobules and adipocyte-like cells consistent with a lipomatous meningioma diagnosis .The tumour cells were positive for epithelial membrane antigen (EMA) and Progesterone Receptor (PgR). Ki-67 was <2%. The patient’s recovery and postoperative course were uneventful. MRI images at a nine-month follow-up show no sign of residual or recurrent disease .She was discharged on postoperative day 7. The neurological examination at discharge was normal except for persistent transient flashing white lights. For this reason, the patient was subjected to the MIDAS-score assessment during the follow-up. The MIDAS-score result was significantly improved with respect to the pre-operative assessments. At one year follow-up, the patient was free of migraine episodes with seldom white light spots which no longer led to difficulty with daily activities.Lipomatous meningioma is a type of meningioma characterized by the presence of mesenchymal cells that are capable of differentiating into different subtypes, including adipose-tissue-containing cells . This acThere are extra-axial lesions with dural tails which usually appear hypodense on CT scans due to the presence of fat , while oImmunohistochemically, cells of lipidized meningioma are reactive for EMA, Vimentin, CD99 and S-100 protein, and on the contrary, it tests negative for GFAP .According to a recent literature review of lipomatous meningioma, there have been about 65 cases of this type  that hadHeadaches can be considered one of the most frequent clinical manifestations of the presence of intracranial masses. Interestingly, an imaging-analysis investigation is more commonly performed for patients presenting with a new onset of a headaches than those with chronic headaches. As for patients with a history of chronic headaches, Whang et al. reported that about 3.7% of these patients have abnormal radiological findings . In addiOur reported case is the first case of a lipomatous meningioma presenting with a history of migraines with visual auras. Based on these findings, various hypotheses arise that can account for the observation regarding the cause of the migraine: there may be traction on vascular structures, compression on the cranial nerves, a neurogenic inflammation through vascular afferents to the meninges or on the cranial vessels ; howeverLipomatous meningioma differs from other meningiomas due to its peculiar clinical, radiological and immunohistochemical characteristics. Seizures and headaches can be the primary onsets of symptoms in lipomatous meningioma. Additionally, precise immunohistochemical findings should be correlated with imaging features that help in the earlier identification of the tumour. A defined diagnosis of lipomatous meningioma is a crucial factor in the choice of surgical intervention. In conclusion, from our case reported here, we observed that the complete resection of the meningioma  led to the resolution of migraines with auras, and after one year the patient presented with a significant improvement in daily life activities.


Sialolithiasis is one of the most common diseases involving the salivary glands. It is a condition that occurs due to an obstruction in a salivary gland or its duct due to a calculus. The formation of a salivary stone is believed to be secondary to the deposition of mineral salts around a nidus, which is frequently associated with a recurrent bacterial infection. Patients with submandibular sialolithiasis usually present with acute swelling over the neck associated with pain, fever, and purulent intraoral discharge. The size of the calculus varies from <1 mm to a few centimeters. The frequency of sialolithiasis is relatively common. It is estimated to affect 12 in 1000 of the adult population. However, the occurrence of giant sialoliths, >15 mm in any diameter, is rare. Here, we describe our experience with a case of giant submandibular sialolithiasis measuring 25 mm presenting as a painless submandibular mass. The patient underwent submandibular gland excision followed by a full recovery. Sialoliths are the most common diseases of the salivary glands after mumps. This condition affects 0.1%-1.0% of the adult population, typically between the ages of 30 and 60 years, with a higher incidence in males . Most siSalivary calculi generally consist of an amorphous mineralized nucleus, surrounded by concentric laminated layers of organic and inorganic substances. The organic components of salivary stones include collagen, glycoproteins, amino acids, and carbohydrates. The major inorganic components are hydroxyapatite, carbonate apatite, whitlockite, and brushite . The etiCommonly, a sialolith measures from 1 to less than 10 mm, with a mean size reported as 6-9 mm. Giant sialoliths are rare and classified as those measuring >15 mm in one dimension [The aim of this paper is to present a case of an unusually sized sialolith and a literature review of current thoughts on preferred technique for imaging the salivary gland, as well as various treatment modalities.A 71-year-old male presented with a two-week history of painless right submandibular swelling that was not associated with fever. The patient had underlying hypertension and diabetes mellitus that were regularly treated.On examination, a right submandibular swelling with normal overlying skin measuring 6 × 5 cm that was non-tender, mobile, and firm in consistency was noted Figure , 1B. ThePreoperative blood investigations , electrocardiography, and chest radiographs were normal. Computed tomography (CT) of the neck was performed as part of the preoperative assessment, which showed opacity in the right submandibular gland and duct Figure , 2B, 2C.Commonly, a sialolith measures from 1 to less than 10 mm, with a mean size reported as 6-9 mm. Giant sialoliths are rare and classified as those measuring >15 mm in one dimension [The diagnosis of sialolithiasis can be made on the basis of its clinical presentation and careful examination. It often presents as recurrent bouts of pain and swelling of the affected salivary gland, usually during mealtimes or in response to other salivary stimuli. In this case, the stone reached its current size without any pain. Bimanual palpation of the floor of the mouth may reveal a palpable stone in most cases. A uniformly firm and hard gland also suggests a hypofunctional or nonfunctional gland .Radiological investigations are deemed very useful in diagnosing sialolithiasis. Radiopaque stones (more than 80% of cases) can be visualized on standard mandibular occlusal radiographs. Radiolucent stones or stones deep within the glands can be investigated using sialography. However, sialography is contraindicated in acute sialadenitis or patients who are allergic to contrast. Ultrasonography (USG) with a high-frequency linear transducer (10-13 MHz) is considered the gold standard and first investigation of choice for parenchymal stones. Its sensitivity, however, decreases in ductal and papillary diseases. Andretta et al. proposed sialo-magnetic resonance imaging  as a noninvasive tool used for diagnosing sialolithiasis . Sialo-MThe role of CT has not been discussed much. As this modality is readily available and the procedure is not time-consuming, it has become a standard investigation tool in our center. The locality of the stones can be determined , as well as the size and number of stones present. These findings will facilitate the surgeon to decide on the best operative method for removal. Interventional sialendoscopy can be used as both a diagnostic and treatment tool ,8. HowevEssentially, the treatment goal is to restore normal salivary gland function. Treatment options may be selected based on the clinical presentation, size, and site of the obstructing stone. Small stones can be “milked out” through ductal orifices by means of bimanual palpation with warm heat application and sialagogue administration. Most cases of sialolithiasis respond to antibiotics with simple sialolithotomy . Large sOur case report describes the removal of a giant sialolith that required excision of the submandibular gland. There are various methods for the treatment of sialolithiasis depending on the affected gland and the size and site of the stones. However, whenever possible, one should choose the most conservative method or the treatment most suitable to the specific situation of the patient.


Pseudoacromegaly is a condition characterized by acromegalic physical features without growth hormone excess, for which radiographic observation has seldom been reported. This is a rare case of pseudoacromegaly. Pseudoacromegaly is a condition characterized by acromegalic physical features without growth hormone excess, for which radiographic observation has seldom been reported. This is a rare case of pseudoacromegaly. Physical assessment revealed enlargement of his nose, lips, and eyebrow arches and positive fist sign (A). An X‐ray showed cauliflower‐like tufting of the distal phalanxes of his hands (B), enlargement of the frontal sinus (C: arrowheads), and heel pad thickness of 22 mm (D: two‐way arrow). Serum growth hormone (GH) and insulin‐like growth factor‐I (IGF‐I) were 0.64 ng/mL and 259.00 ng/mL (+2.7 SD), respectively, whereas an oral glucose tolerance test (OGTT) showed significant suppression of GH production (nadir: 0.17 ng/mL). Contrast‐enhanced pituitary magnetic resonance imaging (MRI) showed no tumor (E) (Figure A 64‐year‐old male with medical history of hypertension and past medical history of ureterolithiasis presented with unsteady gait. His height, body weight, and body mass index were 164.5 cm, 74.6 kg, and 27.6 kg/mThe present case had clinical features resembling acromegaly, but an OGTT and MRI showed that there was no GH‐secreting tumor, suggesting pseudoacromegaly. Pseudoacromegaly or acromegaloidism is a rare syndrome characterized by an acromegalic physical appearance or gigantism without GH/IGF‐I axis abnormalities.The authors declare no conflicts of interest.KY: involved in writing the first draft and managed all of the submission process. KO, HH, and KH: contributed to the clinical management of the patient. FO: organized writing the manuscript.Informed consent was obtained from the patient to publish this case report.


Plasmablastic lymphoma (PBL) is an aggressive type of diffuse large B-cell lymphoma. It is most commonly seen in patients with human immunodeficiency virus (HIV) infection and other immunodeficiencies manifesting commonly in the form of oral lesions.Here, we report a case of an HIV-negative, immunocompetent elderly male who presented with a painful solitary tender lesion on the right anterior lateral thigh. A preliminary diagnosis of osteomyelitis (right femur) from a possibly infected dynamic compression plate was made following initial ultrasound and MRI of the right lower extremity. An attempt was made to incise and drain the lesion, which was abruptly stopped as it resulted in drainage of copious blood, leading to hemodynamic instability. Histopathology of the specimen revealed findings consistent with PBL. The diagnosis of PBL was further confirmed by immunohistochemical staining, which was positive for CD138, MUMI, and CD56 and negative for CD20 and ALK. Due to its rarity and heterogeneous presentations, PBL could be easily overlooked clinically in immunocompetent patients. Therefore, our case highlights the importance of considering the diagnosis of PBL even in lesions whose course is consistent with other infectious bone pathologies. Plasmablastic lymphoma (PBL), according to the World Health Organization (WHO) classification 2016, is characterized by diffuse proliferation of large neoplastic cells morphologically resembling B immunoblasts with the immunophenotype of plasma cells . Due to While HIV-associated PBL accounts for most PBL diagnoses, there are increasingly reported cases in an otherwise healthy population. Most commonly, it presents with mucosal oral lesions . HoweverA 58-year-old male patient presented to the surgical outpatient clinic with undocumented high-grade fever and painful right thigh swelling for one month. He has a history of diet-controlled diabetes mellitus and a motor vehicle collision (MVC) in 2003 and open reduction and internal fixation of the right femur. According to the patient, he was in his usual state of health until one month ago when the swelling, which now involves almost one-fifth of the thigh, started growing rapidly. He also reported associated high-grade fevers and persistent sharp, non-radiating thigh pain, which would aggravate with ambulation.Physical examination revealed a swelling measuring 10 x 15 cm on the anterior lateral aspect of the distal thigh with mild restriction of knee joint movement. The overlying skin was red, warm, and tender to touch without any other significant skin changes. The remainder of the examination was unremarkable. Complete blood count revealed an elevated count of White blood cells  and platelets . The right femur x-ray showed dynamic compression plates (DCPs) and surrounding hyperlucency Figure , 2b. A pIncision and drainage were abruptly stopped as the initial incision resulted in copious blood loss causing hemodynamic instability. The patient was stabilized with nine units of packed red blood cells, four units of platelets, and four units of fresh frozen plasma along with 1 gram of calcium gluconate. Tissue samples were taken for culture and sensitivity and histopathology. After the unsuccessful procedure, the patient’s pain improved, but the blood count continued to suggest an infective process, so empiric antibiotics were started.The histopathology report revealed a tumor comprised of sheets of atypical cells having vesicular nuclei and eosinophilic cytoplasm. At loci, the cells showed prominent plasmacytoid appearance and areas of necrosis. Immunohistochemical staining of the specimen was positive for CD138, MUMI, and CD 56 and negative for CD20 and ALK. CT Pelvis with contrast (Figure PBL is an aggressive form of diffuse large B-cell lymphoma. It is characterized by gradual expression of transcription factors associated with the plasmacytic differentiation, CD38, CD138, MUM1, Blimp1, and XBP1, with decreased expression of CD20 and PAX5 . AlthougOnly two cases of PBL presenting as a soft tissue mass in immunocompetent patients have been published so far ,12. Our Our case highlights the importance of adequate histopathological evaluation for accurate diagnosis and appropriate management of PBL. Physicians should keep in mind the possibility of this diagnosis in such cases. In summary, our case confirms the paramount role of early clinical suspicion of PBL and correct diagnosis in choosing the most appropriate treatment.PBL diagnosis requires a high degree of suspicion and attention from both general physicians and orthopedic surgeons. Because PBL is an aggressive NHL, delayed diagnosis can negatively impact both treatment and survival. Therefore, in particular, orthopedic surgeons should be aware of the varied presentation of this disease, which is often mistaken for soft tissue abscess, osteomyelitis, or an infected implant, leading to a delay in the most appropriate diagnostic evaluation and treatment.


A complete penoscrotal transposition (CPST) is a very uncommon congenital maldevelopment that is always associated with other abnormal body variations . We report a rare case of a term neonate delivered with CPST, which had a flaccid penis and an intact scrotum with unilateral limb and digital deformity, imperforate anus, cardiac malformation a facial deformity. Neonate died two hours after delivery. The foetal abnormality was not detected through routine antenatal services received by the mother. There is a rare occurrence of congenital scrotal anomalies categorized as penoscrotal transposition (PST), bifid scrotum, ectopic scrotum, and accessory scrotum . This reThe aetiology for the abnormal embryological sequence responsible for PST is still unclear , 4. The Complete penoscrotal transposition (CPST) is frequently characterized by major and often life-threatening associated anomalies involving the urogenital, cardiovascular, gastrointestinal, and skeletal systems . Common We attended to a 21-year-old gravida one para zero woman who was referred to Margaret Marquart Catholic Hospital, Kpando, Ghana, on account of the prolonged first stage of labour with poor progress. A 2.4 kg newborn male was delivered at a gestation age of 41 weeks 3 days by a first-trimester scan and last menstrual period (LMP) via caesarean section on the account of nonassuring foetal heart rate. On physical examination, he was noticed to have a complete transposition of the external genitalia with cryptorchidism, hypoplastic penis from the perineum just above the blind anal position, and caudal to the scrotum . Furtherth-week gestation did not indicate any anomaly. Throughout the pregnancy, she was on routine antenatal medications prescribed at the primary attending clinic, and she had no chronic illness. There was no evidence of the mother being exposed to teratogens during pregnancy.The APGAR scores were 2 at the first and fifth minutes. Neonate died two hours after delivery. Other externally associated abnormalities included a laterally rotated left lower limb, which had four digits with a cleft between the third and fourth digits and low set ears. The mother had a booking visit to a hospital at the 11th-week gestation and a regular attendant for antenatal reviews at a primary care facility. Routine laboratory investigations during antenatal care (ANC) such as blood count indices, blood grouping, urinalysis, stool examination and microscopy, fasting blood sugar, retroviral status, and blood film for malaria parasites results were done and essentially normal. Record of the only ultrasound scan taken at 11The woman's 35-year-old husband has a 4-year-old daughter from a previous relationship who was delivered with an imperforate anus. History indicated that both parents of the 4-year-old daughter drink alcohol. Thus, the mother, a housewife, and the father, a driver of a local alcoholic beverage company, drink local alcoholic bitters with the intent to boost their appetite before and during the pregnancy period. However, the mother of the case being reported is a trader that do not drink alcohol. The 4-year-old daughter had a successful surgical reconstruction and currently living a normal life.Penoscrotal transposition (PST) is a congenital urogenital anomaly described first in 1923 by Appleby . Its manth week, the external genitalia are indifferent in both sexes. Under the influence of androgens, the penis and scrotum develop and attain their final position with full differentiation of the organ at the end of the 12th week. The embryological sequence responsible for malformations involving the genitourinary largely remains unclear.CPST is a rare heterogeneous condition that could result from abnormal genital tubercle development around the 6th week of gestation. It could also be associated with a delay in the midline fusion of the urethral folds . Human sComplex genitourinary positional anomalies are often associated with other systems abnormalities that may present as life-threatening during pregnancy, delivery, or postnatal.Previous investigations have reported that anomalies associated with PST such as chordee and hypospadias could be found in 90% of patients, whereas imperforate anus, cardiologic, and gastrointestinal anomalies may be found in 30–60% of victims , 9.The lessons we learnt from both the major and minor reported cases of varied combinations of these organ defects are a mostly functional and aesthetic outcome from surgery ; howeverClinical examination, prenatal obstetric imaging by skilled sonographers/radiologist using standard ultrasound scan machines as a start, and magnetic resonance machines or other modalities of investigations for the purpose enable the identification, classification of degree of anomaly, and monitoring of the progress of these pregnancies. This aims at offering intervention when necessary as well as planning for an appropriate and safe delivery route. Unfortunately, most antenatal clinics currently either lack these skilled labours, the equipment, or both in most developing countries including Ghana to detect organ and skeletal malformations.A case of complete penoscrotal transposition was presented with major or life-threatening anomalies, which were not detected prenatally. This case illustrates the underutilisation of detection in utero as well as organ characterization, which is possible at the first trimester of pregnancy. It also informs us that there is much emphasis on maternal factors in the causes of congenital malformation, with a little investigation on the paternal contributory factors. In this case, the anomalies were incompatible with life and hence required psychological counselling to the parents.It is, therefore, recommended that there is a need for pregnant women to have a complete skeletal scan before the end of the first trimester. There should also be in-depth psychological counselling on postdelivery trauma to parents of babies with such anomalies and perinatal death.


Pyonephrosis is a suppurative infection of the kidney caused by ureteral obstruction. It can lead to kidney failure, septic shock, and death. Thus, it requires prompt assessment and appropriate management. We report a case of a 63-year-old male with giant pyonephrosis contained 10 liters of pus and spontaneously ruptured in the adjacent muscles. This clinical case illustrates the value of computed tomography scan in the diagnosis and management of an uncommon upper urinary tract infection and its complications. Pyonephrosis is a rare and severe urinary infection caused by high urinary tract obstruction. It can result in permanent impairment of renal function, septic shock, or death if it left undiagnosed and untreated A 63-year-old patient without any medical history received for abdominal distension with right flank pain, and fever evolving for 2 weeks. Clinical examination revealed an infectious syndrome, abdominal tenderness with a right flank mass. In addition, there was an inflammatory swelling of the right thigh. Abdominal and pelvic contrast-enhanced CT showed an extensive collection with thin septa, extending from the right hypochondrium to the pelvis, measuring approximately 362 mm in height, 120 mm in transverse diameter and 162 mm in anteroposterior diameter . The rigPyonephrosis is a suppurative infection of the upper urinary tract and differs from infected hydronephrosis by the presence of pus. It is usually associated with parenchymal damage, chronic pyelonephritis and subsequent loss of kidney function The most common clinical presentation is flank pain with fever and chills. Other possible symptoms are nausea, vomiting, back pain, abdominal distension and nephromegaly ,4.Imaging is very essential for the diagnosis and management particularly sonography and computed tomography. Indeed, CT is very sensitive for diagnosis of pyonephrosis and its complications. Moreover, it helps assess renal function, and the perirenal environment looking for perinephric abscess . CT can Giant pyonephrosis has an exceptional anatomical presentation. Rare authors describe huge lesions measuring around 30 cm in the longest axis ,8,9. RupGiant pyonephrosis is a rare suppurative infection of the urinary system. CT plays a key role in the management by determining the etiology and the associated complications. The rupture in the adjacent muscles is an exceptional complication.Have been obtained.


We demonstrate a rare case of inferior mesenteric artery arteriovenous malformations leading to ischemic colitis in a 76-year-old female. Our patient presented with three months of nausea, vomiting, and diarrhea. Colonoscopy displayed diffuse mucosal vascular congestion while CTA and MRA displayed AVMs in the region of the IMA; however, cohesive clinical agreement on AVM from multiple specialties was difficult given its rare occurrence and nonspecific clinical, histopathologic, and directly visualized findings. The three noted dominant AVMs were eventually selected with coil and liquid embolization with successful cessation of symptoms and no major complications. Our discussion focuses on intervention and stressing the importance of radiologic findings, as IMA AVMs, rarely present as ischemic colitis and therefore can clinically masquerade as other etiologies. Ischemic colitis comprises over half of gastrointestinal ischemic insults and is most commonly noted in females, particularly over the age of 60 and with multiple comorbidities . The mosCurrent literature supports the use of MDCT angiography for initial evaluation of inferior mesenteric AVMs with subsequent colonoscopy in order to histopathologically exclude any masquerading disease . DistribTreatment is typically case-specific necessitating diagnostic consensus and clinical cohesion amongst various specialties including gastroenterology, vascular surgery, interventional radiology, and diagnostic radiology. Literature supports arterial embolization at the arteriovenous interface given its efficacy, noninvasive approach, and low complication rates , 8. AthaOur patient is a 76-year-old female with a past medical history of hypertension, type 2 diabetes mellitus, hypothyroidism, and gastroesophageal reflux disease, initially presenting to the emergency department (ED) with a three-month history of nausea, vomiting, and occasional diarrhea. She was evaluated by gastroenterology, and an outpatient EGD was performed showing a small hiatal hernia and atrophic gastritis. Within the next few weeks, she presented to ED with acute onset abdominal pain and worsening diarrhea which now appeared to be bloody. A CT scan of abdomen and pelvis was requested for the clinical diagnosis of ischemic colitis. Significant colonic wall thickening and pericolonic inflammatory stranding of the entire colon with sparing of the cecum, concerning for pancolitis with mild ascites, were noted on the initial CT scan Figures . The gasApproximately one month later, the patient presented to the emergency department with bilateral lower extremity edema and fatigue. Work-up for the edema revealed bilateral lower extremity DVTs, and the patient was placed on Lovenox. Shortly after, the patient developed several episodes of hematochezia and abdominal pain which prompted MRA of the abdomen, further strengthening the concern for AVMs. The MRA exhibited an inferior mesenteric artery distribution of multiple focal aneurysmal vessels in the left colonic mesentery Figures . AdditioAt this point, interventional radiology was consulted for a mesenteric angiogram. Arteriogram demonstrated a total of 3 AVMs, each individually supplied by branches of the IMA Figures . The lefDuring postprocedural follow-up, the patient denied any abdominal pain, melena, or hematochezia. The patient demonstrated stability for over 1 year and was without any recurrence of abdominal symptoms.This is a novel case for several reasons and elucidates the importance of utilizing multiphasic cross-sectional imaging for an accurate diagnosis given the rare presentation. As previously noted, IMA distribution of AVMs is the least common distribution of splanchnic vascular anomalies. Furthermore, instead of presenting with the more common portal hypertension-associated symptoms, our patient presented with the less common presentation of ischemic colitis with hematochezia and diarrhea.Initial confusion in diagnosis occurred for several reasons in our case. Retrospective evaluation of the initial ED non-IV contrast CT abdomen and pelvis did display tortuous vasculature in the IMA distribution; however, given that no IV contrast was given, this finding can easily be overlooked. Colonoscopy showed mucosal vascular congestion, and histopathology revealed architectural distortion seen in chronic colitis; thus, an acute ischemic colitis was considered less likely. The subsequent CTA of the abdomen and pelvis did however delineate the vascular malformations very well. However, given the rarity of AVM-induced ischemic colitis and pathology findings, clinical management of chronic colitis was continued. This highlights the importance of scrutinizing the associated intestinal vasculature when bowel pathology is demonstrated. Multimodality multiphasic cross-sectional imaging (CTA and MRA) built a very strong diagnosis of IMA distribution AVMs, allowing visualization of the niduses in the arterial and venous phase along with any underlying vascular pathology that could preclude intervention. Mesenteric angiogram further confirmed the suspected three niduses in the IMA distribution. Additionally, the angiogram allowed the interventional team to deem the AVMs amenable to liquid and coil embolization as they did not appear too high flow or large for intervention.This case report is limited due to the lack of postprocedural imaging which would allow for thorough evaluation of the vascular anatomy and collateralization, serving as a beneficial baseline if future complications were to arise. However, in our case, the patient had no complications in the postprocedural course, and therefore, imaging was not clinically indicated. The multimodality imaging of this case report  allows for wholesome diagnostic and interventional evaluation and augments our understanding of a rare vascular anomaly.Ischemic colitis secondary to AVMs can be strongly suggested and diagnosed on imaging, and with multispecialty diagnostic consensus can lead to early treatment and fewer complications. This elucidates that the imperative role of radiology can play in suggesting a firm diagnosis and providing appropriate intervention.


This study aimed to review the surgical excision results and pathological diagnostic features of rarely observed intracardiac masses in the light of the literature. Diagnosis and treatment approaches and complications were evaluated.Forty patients , who had undergone surgery for intracardiac mass between January 2008 and December 2018, were included in this study. The patients’ data were analyzed retrospectively from the medical records of both centers.When the pathological diagnoses were examined, 85.8% of the masses (n=35) were observed to be benign (benign tumor + hydatid cyst) and 14.2% (n=5) were malignant tumors. The masses were most commonly located in the left atrium , and this was followed by the right ventricle , right atrium , and left ventricle . Of the patients, 7.5% (n=3) died during the early postoperative period, while the remaining 92.5% (n=37) were discharged with healing. In the histopathological diagnosis of the patients, in whom in-hospital major adverse cardiovascular events were observed, there was malignancy in two cases.Intracardiac masses, which have pathological features, are severe life-threatening problems. In-hospital mortality is frequent, especially in malignant tumors. Although benign and malignant cardiac tumors are rare, they are important causes of mortality and morbidity. Cardiac neoplasms are asymptomatic until they reach a large size, and present with nonspecific findings. Like myxomas, the origin of some of these tumors is uncertain. Cardiac masses have become a surgically treatable pathology with the introduction of the heart-lung pump. Malignant tumors of the heart could not get rid of the poor prognosis despite all advances.. In another autopsy series, from Germany, this rate is also shown in the range of 0.001-0.3%. Primary tumors of the heart may be benign or malignant. Secondary tumors are metastatic and malignant tumors. Of all cardiac tumors, 75% are benign and 25% are malignant. Myxomas are the most common primary cardiac tumors. In this study, we aimed to present the incidence, localization, histopathological diagnostic characteristics, and surgical approaches of intracardiac masses.The incidence of cardiac tumors is observed to be 0.02%, based on 22 large autopsy data from a United Kingdom centreA total of 40 patients , who had been operated in the Erciyes University Faculty of Medicine and Kayseri City Hospital Cardiovascular Surgery Departments, between January 2008 and December 2018 and whose postoperative tissues had been sent to the pathology clinic, were included in the study. The study was designed as a retrospective record review study. The approval of the local ethics committee was obtained.All patients underwent parasternal long and short axis and apical four-chamber transthoracic echocardiography (TTE) (Vivid 7 GE) before the procedure. TTE was the common preoperative diagnostic method used in all patients for the location, size, and general characteristics of the mass. If necessary, transesophageal echocardiography (TEE) was performed under local anesthesia using a Vivid 7 GE device.All patients underwent surgery under general anesthesia. A median sternotomy incision was performed in 39 patients. Only in one patient, diagnosed pathologically with myxoma, the mass was removed by preferring the right anterior thoracotomy incision. Following the classic median sternotomy, aortic cross-clamp and standard cardiopulmonary bypass and antegrade cardioplegia were performed together with aortic and bicaval venous cannulation. The incision performed for tumor resection was selected by considering the location of the tumor.In hydatid cyst cases, saline-impregnated gauze was placed around the cyst so that the cyst content was not spilled outside. Some of the cyst content was aspirated with a syringe, and 3% serum sale was injected into it, and then the cyst content was carefully removed, and the cyst was cleaned. After this procedure, cystectomy + capitonnage was performed in all patients. While the defects formed after the removal of the mass were closed with primary sutures, the defect created after resection was closed by polytetrafluoroethylene in one patient with lipoma in the right atrium and by using a pericardial patch in two patients.Statistically, the distribution of quantitative data was defined as x±standard deviation. The difference between the two groups was evaluated by Student’s t-test. Qualitative numbers were defined as percentage. The statistical significance level was set at 0.05.A total of 40 patients  were included in the study. The baseline characteristics of the patients are presented in While left atriotomy was the preferred heart chamber in 19 cases, the transseptal incision was used to remove the mass in eight cases .And although 62% of the cases were myxomas  and B, 1By considering the location of myxomas in the heart chamber, while the right and left atrium were opened separately together in two cases, the transseptal intervention was preferred in four cases. The created defects were repaired using a primary or pericardial patch. In one case, the mass in the left atrium was removed by preferring the right anterior thoracotomy incision.Surgical interventions, saphena-posterior descending artery, and LIMA-LAD anastomoses were performed concomitantly in one patient in whom myxoma was excised in the right ventricle.In another patient with left atrial myxoma, mitral valve replacement was performed at the same session upon observing the third mitral insufficiency after the intraoperative TEE.Again, after the resection of the existing mass in the right atrium, a 4 x 4-cm mass in the right lung basal lobe posterior was removed by wedge resection during the same session. Both masses were diagnosed as a hydatid cyst.In another case, who presented with ischemia in the left lower extremity and whose left atrial mass was diagnosed as myxoma, thrombectomy was performed in the left lower extremity. The patient presented with a mass located in the left atrium diagnosed as a hydatid cyst, which is a nontumoral lesion in the pathology  and B, aIn another case reported as undifferentiated angiosarcoma  and B, tIn another patient, who was six-month-old, perforation was detected in the region of the ileum close to the cecum during the postoperative period. The last part of the ileum, the cecum, the appendix, and the 5-cm segment of the colon were removed. The patient, in whom ileostomy and colostomy were performed, died in the early postoperative period.Another patient, who was diagnosed with squamous cell carcinoma metastasis, was re-hospitalized on the eighth postoperative day due to the development of sternal infection, and the patient to whom sternal repair was applied was discharged with treatment.The removed mass was diagnosed as glandular in three of our patients with myxoma and as a cellular variant in one of the patients. In all of these patients, it was noted that the location was the left atrium.The number of early mortality cases in the postoperative period was three (7.5%), two of whom were from the malignant mass group.-5; they make up about 50% of those and are observed more frequently in women of middle age.Cardiac tumors are rare and account for 0.2% of all tumors in humans. These are divided into two types, as primary and secondary/metastatic tumors. Secondary or metastatic tumors are 20-40 times more common than primary tumors. Primary intracardiac tumors are rare. Of the tumors, 75% are benign. Cardiac myxomas represent the most frequently observed primary cardiac tumors. Myxomas are often observed as a single mass with a spherical shape and smooth surface originating from the left atrium endocardium, adjacent to the foramen ovale. However, macroscopically, they may have fingerlike protrusions, an irregular surface, and, sometimes, the appearance of a bunch of grapes. While 21 (84%) of the myxomas were located in the left atrium, two (8%) were in the right atrium and two (8%) in the right ventricle. Myxomas are a rare cause of acute peripheral arterial ischemia. In our study, 60% of myxomas were detected on the left atrium side in accordance with the literature, and the majority were interfered with left atriotomy.Myxomas were reported to be in the left atrium in 75-80% of the cases, in the right atrium in 10-20% of the cases, and in the atrium and ventricle in 5-10% of the cases-10.Myxomas may sometimes present with lower extremity embolism as the first symptom in the clinic. Among our cases, three patients were diagnosed with a glandular myxoma, and one was diagnosed with a cellular myxoma. It was remarkable that these four cases were located in the left atrium. Those, which are evaluated as a glandular myxomas, should be differentially diagnosed with metastatic adenocarcinomas. Myxomas are easily diagnosed pathologically with their typical histopathological appearance and classical locations. Although there is no statistically significant difference in myxomas in terms of glandular differentiation and prognosis, recurrence or metastasis has been reported in the literature. In a study, when the deoxyribonucleic acid was examined by the polymerase chain reaction method in tumor tissues after surgery, most of the cardiac myxomas were observed to be polyclonal. Their polyclonal origin characteristics may contribute to tumor recurrence.In the literature, there are studies showing that clonality may play a role in myxoma recurrences. In all of our myxoma cases, during the excision of the mass, care was taken to remove it together with some part of the heart chamber where it was located. In the treatment of these patients, there was no need for additional treatment, and cure was provided with surgical treatment.The treatment of myxomas is the surgical removal of the mass under cardiopulmonary bypass. The outcome of surgical treatment was excellent. Cardiac echinococcosis is rare and occurs in 0.5-3% of all cases,14. Clinical presentations are directly related to the location, size, and integrity of cysts and may vary from asymptomatic to sudden death. In the microscopic examination, typical eosinophilic lamellar, with cuticular membranes, scolices are observed and can be easily pathologically diagnosed as macroscopic and microscopic. Even though the cardiovascular hydatid cyst is rarely observed, its early diagnosis and surgical management are essential. In our cases, it was diagnosed echocardiographically as cystic lesions and was immediately operated. As a result, successful resections were made, and mortality was not observed. In our study, 12% of total cases formed hydatid cysts and most of these cases were detected in the left system.In the nontumoral hydatid cyst lesion, which is one of the causes of a mass in the heart, the most affected regions are, respectively, the liver, lung, brain, spleen, kidney, bone, muscle, pelvis, heart, spinal cord, and retina. The case of lipoma in our series was successfully resected, and no complication developed.Cardiac lipomas are observed rarely and mostly consist of benign tumors, frequently remaining asymptomatic during a patient’s lifetime. These tumors are usually located in the epicardium and cardiac cavities. Interatrial lipomatous hypertrophy found in the pathological differential diagnosis is a rarely observed non-capsular hamartomatous lesion and is also called as lipomatous hypertrophy of the atrial septum. Even though the resection of cardiac lipomas is mostly easily performed, a small number of patients with extensive spread, unfortunately, undergo a difficult surgery, having poor outcomes. A cardiac hemangioma may emerge at every age, in any localization, such as the pericardium, endocardium, or the myocardium in the heart. The histological subtypes of cardiac hemangioma are as follows: cavernous hemangioma, capillary hemangioma, and arteriovenous hemangioma. The cavernous hemangioma consists of multiple dilated vessels with thin-thick walls. Surgery is the best way to treat cardiac hemangioma. The prognosis after surgery is good.Hemangioma of the heart, which presents as a primary cardiac tumor, is sporadic and constitutes about 2% of all primary cardiac tumors. They are well-circumscribed, solid masses, which are either grown into the cavity or embedded into the myocardium and they are often numerous. From a histological aspect, every mass is composed of large cells with clear cytoplasm-spider cells, which take their name due to their radial cytoplasmic strands. Rhabdomyomas are spontaneously regressing tumors at a rate of 50-100%. Therefore, clinical follow-up is recommended for asymptomatic patients. One of our cases was a case of rhabdomyoma located in the left atrium.Rhabdomyomas, which constitute 60-86% of fetal cardiac tumors, are associated with tuberous sclerosis at a rate of 52-79%. The tumor is considered to be a hamartoma instead of a true neoplasm. The most common sarcomas are angiosarcoma and undifferentiated pleomorphic sarcoma. Angiosarcoma, the most common type of sarcoma, is located most frequently in the right atrium in all age groups. The pericardium is frequently involved. Cardiac tamponade and pericardial effusion are common complications. They are often not well-circumscribed, hemorrhagic, and appear as necrotic masses. Histopathologically, the vascular channels formed by atypical, pleomorphic endothelial cells attract attention. Occasionally, the areas of hemorrhage and necrosis and mitotic figures accompany them. Histopathology analyses must contain immunohistochemical (IHC) stain, and a large panel of antibodies should be carried out for the purpose of avoiding misdiagnosis. Angiosarcomas are positive for vascular markers, including cluster of differentiation (CD) 31 and CD34. Most tumors are poorly undifferentiated. Cardiac angiosarcomas frequently have a poor prognosis, even with treatment. They usually affect men at the age of 40 to 50 years, and they have a worse prognosis in patients at a younger age. In our study, a patient diagnosed with undifferentiated angiosarcoma was examined and the tumor proliferation index was found to be high in Ki67 IHC staining. Twenty days after the complete removal of the mass by operation, the patient applied to cardiac surgery, and the mass was cleaned again through reoperation. However, the patient died due to intracranial hemorrhage despite all efforts.Malignant heart tumors originate from the myocardium, endocardium, epicardium, and pericardium. The pericardium is frequently the localization of metastases. Of the malignant tumors, about 75% are sarcomas.Cardiac leiomyosarcoma constitutes less than 1% of cases. The median age at diagnosis was 48 years with a female predominance. Cardiac leiomyosarcomas are most frequently located in the left atriumIn the pathological diagnosis, the tumor was observed to be of blunt, fusiform appearance, and as bundles constituted by cells, of which cytoplasm boundaries were not clearly selected. Necrosis, hemorrhage, and myxoid degeneration areas may be observed between the tumor cells. Meanwhile, pleomorphic cells, osteoclast-type giant cells, and increased mitotic activity may accompany them. In IHC examinations performed for differential diagnosis, while vimentin, smooth muscle actin, myoglobin, and positive reaction in tumor cells are expected, a negative result is obtained with factor VIII-related antigen, epithelial membrane antigen, and CD34.Leiomyosarcomas are tumors with a poor prognosis and have a frequent recurrence and distant metastatic potential. The primary treatment is the complete excision of the mass with a clear surgical margin, and with the multidisciplinary approach, chemotherapy and/or radiotherapy can be used palliatively in some cases. In our series, the diagnosis is leiomyosarcoma, located in the left ventricle in a 39-year-old male patient. The patient was evaluated at the oncology council and chemotherapy was planned. After the extensive surgical excision of the mass, the patient died before starting chemotherapy.,26. It is usually of a non-Hodgkin type and includes only the heart and pericardium. Although more than 20% of the patients with nodal, extra-nodal, disseminated lymphoma may have cardiac involvement, it may also be observed as lymphoproliferative diseases in immunosuppressive patients. Approximately 80% of primary cardiac lymphomas in immunocompetent hosts are diffuse B-cell lymphomas, and those are found in subjects with immunodeficiency states. The right atrium and right ventricle are their frequent localizations. Clinical presentation is heterogeneous and is usually associated with the site of involvement in the heart. Histopathological diagnosis is the main diagnostic method, and typification requires a large IHC staining panel such as CD20, CD5, CD3, CD10, B-cell lymphoma 2 protein, and Ki67. Chemotherapy has been utilized alone or in combination with radiotherapy. Palliative cardiac surgery has been carried out, primarily for the purpose of tumor debulking. The combination of chemotherapy and radiation therapy is accepted to be the treatment of choice. The survival is usually less than a month without treatment,26. In the series we presented, there are two malignant B-cell lymphoma cases. Two patients died without chemotherapy.Primary cardiac lymphoma represents a malignancy, which is observed very rarely,5. In the presence of metastatic tumors in the heart, metastatic foci are also usually found in other localizations. In very rare cases, the heart is the first and only metastatic focus. Pericardial involvement is frequently observed. Lung, esophagus, melanoma, breast, renal cell carcinoma, choriocarcinoma, and childhood rhabdomyosarcomas are the most common metastatic tumors in the heart. Head and neck cancers rarely cause distant metastasis, involving the heart. In our series, there is a case of squamous cell carcinoma, of which the primary region is the head and neck region.Metastatic tumors in the heart are more commonly observed than primary tumors. Metastatic tumors to the heart are observed at least 100 times more frequently in comparison with primary cardiac tumors when identified at autopsy, occurring in 1-3% of all autopsied patientsThe most important limitations of this study are the small number of patients and the small number of centers.Intracardiac masses, which have pathological features, are severe life-threatening problems.It is inevitable that masses in the heart, which is a vital organ, will be treated as privileged without making a benign-malignant distinction. The signs and symptoms of cardiac masses depend more on their localization than a histological diagnosis. In principle, patients who are eligible for surgery are treated urgently with the risk of sudden death and embolism in mind. In particular, malignant tumors involve severe mortality.


We describe the case of a female patient with calcification in renal topography, initially diagnosed as lithiasis in the left kidney, and later attributed to calcification of intrarenal vascular aneurysm. Next, we discuss the relevance of considering such an entity in the differential diagnoses of intrarenal calcifications before choosing any form of specific interventional treatment. A wrong diagnosis can compromise treatment and even lead to complications. This report illustrates this situation, in which the wrong diagnosis could have evolved catastrophically, and reinforces the alert to the need for attention to the quality of the exam, and analysis of the report. In case of doubt, ratify with exams that are more accurate.Urinary lithiasis is a prevalent and recurrent disease, whose diagnosis is usually easily made by means of radiological investigation. The definition of which modality should be used is made by analyzing the characteristics of the calculus, patient, and the known results of the various intervention methods.A 71-year-old woman was referred for urological counseling due to the finding of urinary calculus in a routine ultrasound examination. Patient was asymptomatic and with no past history of urinary lithiasis. Her hypertension was controlled with regular use of candesartan cilexetil and hydrochlorothiazide.She presented with recent and normal laboratory tests . Ultrasonography of the urinary tract showed topical kidneys with normal dimensions and a 7.5mm calculus in the middle third of the left kidney, with no dilatation of the urinary tract . Right kDespite being asymptomatic, she requested that some form of treatment be considered because of her concern about developing symptoms due to calculus migration and the need for emergency treatment.As part of the treatment planning, a computed tomography scan was performed (without contrast), which showed an unchanged right kidney. The left kidney was unaltered, but the images suggested a saccular aneurysm with parietal calcification of the left renal artery in the plane of the renal hilum, measuring 10mm x 10mm  and 3. UFaced with the unexpected finding, and diagnosis of urinary calculus ruled out, she was referred to a vascular surgeon. Renal artery computed tomography angiography showed that the right kidney was supplied by two arterial branches of similar caliber, patent throughout, without stenosis or aneurysm. The left kidney was supplied by a single arterial branch, with a prominent aneurysm involving the anterior segment branch, showing almost complete thrombosis, measuring 12mm x 9mm in diameter . The diaThe patient remains guided by the vascular surgeon only for routine follow-up and intensive control of blood pressure levels.Faculdade de Medicina do ABC under # 4.440.144, CAAE: 34350420.6.0000.0082.This study was approved by the Research Ethics Committee of -3Renal artery aneurysm is an uncommon condition, with an incidence of 0.01% to 2.5% in the population, and an average growth rate of 0.06mm to 0.6mm per year. It predominates in females (72%) during the sixth decade of life. Symptomatology is poor and usually quite rare, consisting of abdominal flank pain and hematuria. It may course with hypertension, palpable and pulsatile renal mass, renal artery murmur, calcification, and thromboembolism. Its treatment is controversial, and in most cases it is conservative. However, endovascular interventional treatment may be indicated in case of rupture, or when it is understood that the risk of this event is high.Sataa et al. reported a similar case, in which the aneurysm was misdiagnosed as renal lithiasis on ultrasound and radiography, leading to inadvertent treatment with one session of extracorporeal shock wave lithotripsy, without complications. In this case, the diagnosis of aneurysm was determined after computed tomography scan investigation in preparation for the second session. John demonstrated the case of a patient with calcification of bilateral renal artery aneurysm.There are some similar case reports of renal artery aneurysm in the literature. Rafailidis et al. reported the case of a 72-year-old patient with a renal artery aneurysm diagnosed as an ultrasound finding.The particularity in the reported case is the striking similarity of the calcified aneurysm with renal lithiasis on screening imaging. The fact that the calcified aneurysm was located in the intraparenchymal segment of the renal artery led to diagnostic confusion. A computed tomography scan to define the treatment determined a radical change in diagnosis and management. Extracorporeal shock wave lithotripsy, if chosen without recognition of the real diagnosis, could have been accompanied by hemorrhagic complications, which could have put the patient’s life at risk. This case offers some considerations on the radiological investigation in the preparation prior to the intervention. The suggestion is that in selected cases of higher complexity of urinary lithiasis, a more accurate imaging test of the urinary tract should be considered for a better diagnostic and therapeutic definition.


Pancreatic lesions are more commonly identified nowadays with the widespread use of imaging investigations. Pancreatic lesions are heterogeneous groups of pathologies with different behavior and prognosis. It is quite difficult to differentiate these lesions because of the shared clinical manifestation and the overlapping imaging features of these lesions. We report the case of a 38-year-old woman who presented with a complaint of a 3-month history of epigastric abdominal pain radiating to her back. She reported a gradual increase in the severity of the pain. She described it as sharp in quality and was exacerbated with food intake and was alleviated by oral paracetamol. There was no history of abdominal distension, weight loss, or change in appetite. Apart from asthma, she had no other comorbid conditions or previous surgeries. She was referred to have an abdominal computed tomography scan which showed a large lesion that appears to arise from the head of the pancreas and was cystic with no soft tissue component. The radiological impression was a lymphoepithelial cyst, duplication cyst, pseudocyst from previous acute pancreatitis, lymphangioma, or intraductal papillary mucinous neoplasm. The decision for open surgical removal was decided. The patient underwent laparotomy and the cystic lesion was identified as arising from the pancreatic head. Complete resection of the mass was carried out with no complications. Histopathological examination revealed cystic lesions with the presence of lymphoid cells aggregates peripherally. The lining of the channels was positive for CD31 and was negative for CD34. Such findings conferred the diagnosis of lymphangioma. Lymphangioma is a very rare tumor of the pancreas with non-specific clinical and imaging features. Complete surgical resection of the lesion is the treatment of choice and the prognosis of the tumor is favorable if the tumor was resected completely. With the widespread use of imaging modalities, pancreatic lesions have been increasingly identified. In a study involving over 1400 patients who underwent magnetic resonance images of the pancreas, the incidence of cystic pancreatic lesions was approximately 20% . Here, wA 38-year-old woman presented to the general practice clinic with a complaint of a 3-month history of abdominal pain. The pain was in the epigastric area and radiated to her back. She reported a gradual increase in the severity of the pain. At the time of presentation, she scored it as 4 out of 10 on the severity scale. The pain was not awakening her from sleep. She described it as sharp in quality and was exacerbated with food intake and was alleviated by oral paracetamol. There was no history of abdominal distension, weight loss, or change in appetite.The patient was known to have asthma that was poorly controlled mainly due to non-adherence to her medications and non-avoidance of the triggers. She had no other comorbid conditions or any surgical history of any operation. Her routine medications included inhaled salbutamol and corticosteroid. She was a non-smoker and non-alcoholic. She worked as an interior designer. The family history was positive for celiac disease and colon cancer.She was vitally stable with a normal pulse rate, blood pressure, temperature, and respiratory rate. Abdominal examination yielded normal findings with no localized tenderness or guarding. Bowel sounds were of normal intensity and frequency. The patient underwent some laboratory investigation that included C-reactive protein (5.2 mg/dL), erythrocyte sedimentation rate 10 mm/hour), and amylase (80 U/L), which were within the reference range  considering the large tumor size and its location. The patient underwent laparotomy and the cystic lesion was identified as arising from the pancreatic head. Complete resection of the mass was carried out successfully without the need to resect the pancreatic head as the initial plan. The patient had an uneventful recovery. She was discharged 7 days later.The histopathological examination of the specimen revealed cystic lesions with the presence of lymphoid cells aggregates peripherally Figure . The linWe reported a case of pancreatic lymphangioma in a young woman presenting with chronic epigastric pain. Pancreatic lymphangioma is a benign tumor that though to originate from blockage of the pancreatic lymphatic vasculature due to congenital or acquired etiologies . Such acPancreatic lymphangioma is very rare and accounts for less than 1% of all pancreatic lesions -9. PancrWhile the imaging studies may suggest the diagnosis of lymphangioma, histopathological examination is required to establish a definite diagnosis. As in the present case, the lymphangioma exhibit positivity for CD34 . The CD3Fine-needle aspiration via the endoscopic ultrasound guidance has a role in making the diagnosis of pancreatic lesions . HoweverLymphangioma is a very rare tumor of the pancreas. As the present case highlighted, the clinical and imaging features of this tumor are non-specific. Pancreatic lymphangioma should be considered in the differential diagnosis of pancreatic cystic lesions. Complete surgical resection of the lesion is the treatment of choice and the prognosis of the tumor is favorable if the tumor was resected completely.


Mucinous cystadenoma is a benign tumor that is commonly found in the pancreas, ovaries, or appendix, but is rarely encountered in the lungs. Worldwide, only a few reported cases of these tumors originate in the lungs. Herein, we analyzed the imaging features of a case of pulmonary mucinous cystadenoma (PMCA). To the best of our knowledge, this is the first reported case of PMCA complicated by significant infection.A 57-year-old man was admitted to our hospital with blood in sputum for more than 2 months. Serum laboratory examination showed significantly elevated leukocyte and tumor marker, carcinoembryonic antigen. Enhanced thoracic computed tomography and whole-body positron emission tomography/computed tomography showed a cystic-solid ill-defined mass in the right upper lung.The tumor was considered malignant, both clinically and radiologically.The patient underwent right upper lobe tumor resection and mediastinal lymph node dissection.Postoperative specimen pathology was diagnosed as PMCA with infection. The patient was not administered any further treatment. The patient was alive without any recurrence or metastasis of the tumor after 2 years of follow-up.Preoperative diagnosis of PMCA with atypical imaging and clinical manifestations is extremely difficult. This is the first reported case of PMCA complicated by a significant infection that was misdiagnosed preoperatively as a malignancy. They were located in the periphery of any lobe of the lung, with a preference toward the right side.,4 The tumor is usually unilocular and filled with mucus.–5 The cyst wall was lined with mucinous epithelium with varying degrees of atypia.–5 Typical imaging features of this tumor tend to be well-demarcated thin-walled singular cystic masses in the periphery of the lung parenchyma.,5 When the mass is large, it can cause distortion of the surrounding tissue, resulting in inflammation and atelectasis. Herein, we report a rare case of PMCA complicated with infection, which was misdiagnosed as a malignancy on preoperative computed tomography (CT), positron emission tomography/CT (PET/CT), and clinical findings.Mucinous cystadenoma of the lung is a rare benign tumor that has rarely been reported.2A 57-year-old male patient was referred to our hospital because of blood in the sputum for more than 2 months and a lung mass found on a plain CT scan in a local hospital. The patient had a small amount of white mucous sputum with bright red blood filaments, without chest pain or abnormal body temperature.9/L in the blood routine, and neutrophils accounted for 90.6%. In the male tumor markers combination (14 items), carcinoembryonic antigen (CEA) serum level increased to 19.76 ng/mL , and the other markers were within the normal range.The patient's laboratory examination in our hospital showed that the white blood cell count increased to 13.59 × 10Chest contrast-enhanced CT revealed a 53 × 46 mm mass in the posterior segment of the right upper lobe with heterogeneous enhancement, extensive central liquefaction, a few striped shadows around the tumor, and multiple enlarged mediastinal lymph nodes Fig. A to C. LWhole-body PET/CT showed a round cystic-solid mass in the posterior segment of the right upper lobe, with irregular margins. The thickness of the cyst walls was not uniform. The cystic wall showed significantly increased fluorodeoxyglucose (FDG) uptake , but the central cystic component was not accompanied by FDG uptake. Several enlarged lymph nodes with increased FDG uptake were observed in the right hilum and mediastinum, and the maximum standardized uptake value was approximately 4.82  was reexamined, and the results were all within the normal range with a CEA value of 2.31. The patient was alive without any recurrence or metastasis of the tumor after 2 years of follow-up.Ethical approval was not required because this was a case report of the patient's clinical information. Written informed consent was obtained from the patient for the publication of this case report and accompanying images.3 Mucinous cystadenoma is a benign tumor that is commonly found in the pancreas, ovaries, or appendix. Worldwide, only a few reported cases of these tumors originate in the lungs. PMCA was first described by Gower in 1978 as “an unusual mucous cyst in the lung.” The age of patients with PMCA ranges from 32 to 75 years , and the tumor seems to be more common in women. They were located in the periphery of any lobe of the lung, with a preference toward the right side. The median size was 5 cm . Although our patient was male, he was nearly 60 years old, with a mass size of approximately 4.6 × 5.3 cm, also located in the right lung, which was generally consistent with the literature reports.Mucinous cystadenoma is defined as “a localized cystic mass filled with mucin and surrounded by a fibrous wall lined by well-differentiated columnar mucinous epithelium.”–4 Immunohistochemical (IHC) studies usually show positivity for pan-cytokeratin (CK) and, in some cases, CEA, surfactant-associated protein A, and negative stain for TTF-1. These IHC features suggest that the tumor is derived from the non-alveolar epithelium.,4 A positive CEA indicates that the tumor has a local malignancy or is prone to recurrence after surgery. IHC examination of our patient specimen showed positive CK7 and negative TTF-1 expression, which was consistent with the literature reports. Although CEA staining was not performed, the patient's serum CEA level was significantly elevated preoperatively, suggesting that the tumor had malignant potential.The histology of this neoplasm is characterized by a localized cystic mass and benign proliferation of mucus-producing epithelial cells.,4 PMCA is usually well-circumscribed and homogeneous on CT, and the cyst wall is usually thin. Inflammation and adjacent atelectasis only seem to be present after a certain enlargement of the lesion due to compression and distortion of the surrounding tissue.–5 On FDG PET/CT, the tumor usually shows no abnormal uptake due to its thin wall and mucus-filled interior. In our case, the findings on CT and PET/CT were different from those reported in the literature. The interior of the tumor was mainly cystic, but there were still uneven high-density lesions, uneven wall thickening, and obviously abnormal FDG uptake, which led to misdiagnosis as a malignant tumor, but histopathologically confirmed as mucinous cystadenoma with inflammatory changes.Radiographically PMCA appears as a well-defined, homogeneous lesion. Sometimes, depending on the local condition, resection of these lesions can also be minimally invasive. Further follow-up is usually required based on pathological findings. The prognosis for these patients is usually good, with recurrence being rare and metastases from malignant lesions being exceptional.,6,7 Matsuo et al reported a case of PMCA with tumor recurrence 20 years after surgery, which was the only case of recurrence reported in the literature. Matsuo et al reported a case of pulmonary mucinous cystadenocarcinoma arising from PMCA, and Davison et al reported a case of adenocarcinoma arising in a mucinous cystadenoma of the lung. In our patient with PMCA, the serum CEA level was as high as 19.76 ng/mL before surgery, and it was reduced to 2.31 ng/mL 1 year after surgery, which also indicated that this tumor had a certain degree of malignancy and required regular follow-up. KRAS mutations, which are typical for mucinous carcinomas of the lung, may also be a mechanism for the development of mucinous cystadenocarcinoma arising from PCMA.,4Although PMCA is a benign tumor, it may have some degree of malignant potential, and early and complete resection with a sufficient margin is required for the treatment.In conclusion, a mucinous cystadenoma is usually a grossly well-demarcated peripheral cyst filled with gelatinous mucin, but this benign tumor may have some degree of malignant potential. We encountered an extremely rare case of PCMA associated with infection. The CT and FDG PET/CT imaging findings of this patient were relatively special, and the serum CEA level was also significantly increased. The clinical diagnosis was very difficult, and complete resection of the tumor was necessary.Conceptualization: Xun-Ze Shen (Shen XZ).Formal analysis: Zhou-Ye Luo (Luo ZY).Resources: Fang Liu (Liu F).Writing – original draft: Xun-Ze Shen (Shen XZ), Zhou-Ye Luo (Luo ZY), Chen Lin (Lin C).Writing – review & editing: Xun-Ze Shen (Shen XZ).


Desmoid-type fibromatosis (DF) is a rare tumor that develops in the limbs, abdominal wall, and abdominal cavity. It is accounting for less than 3% of soft-tissue sarcomas and less than 0.03% of all neoplasms.A 57-year-old man was diagnosed as acute peritonitis due to intra-abdominal tumor rupture. Since his systematic symptoms were relatively stable, gastrointestinal perforation was ruled out, the differential diagnosis of the tumor itself was difficult, and it was unclear resectable by emergency surgery, we started conservative treatment. After examinations, ileocolectomy was performed. Histopathological examination revealed spindle cells with collagenous fiber hyperplasia and immunohistochemical staining for β-catenin was positive, so we made a diagnosis of mesenteric desmoid-type fibromatosis (MDF).The mechanism of DF development is suggested to be associated with hereditary diseases, mechanical stimuli, and a history of exposure to radiation appear to be involved as pathogenetic factors in sporadic development. Surgical resection is the first-line treatment for MDF, but the postoperative high local recurrence rate is problematic. Drug therapy and radiation therapy are selected for cases in which radical resection is not possible or for recurrent cases. However, the number of examined cases is small and sufficient evidence has not been accumulated for most treatment strategies, it is expected that the optimal treatment at the time of recurrence will be further verified by the accumulation of MDF.There are few reports of peritonitis caused by MDF rupture; emergency surgery can be avoided. •Mesenteric desmoid-type fibromatosis (MDF) is rare tumor, difficult to diagnose because of nonspecific signs and symptoms.•There are few reports of peritonitis caused by MDF rupture; emergency surgery can be avoided.•Surgical resection is the first-line treatment for MDF.•Local recurrence may occur due to tumor rupture of the abdominal cavity, so careful follow-up is essential. It is accounting for less than 3% of soft-tissue sarcomas and less than 0.03% of all neoplasms. The DF frequency of occurrence is 2.4 to 4.3/million. DF is more common among women and is classified into three types: extra-abdominal wall type affecting the head, neck and limbs (43%), abdominal wall type (49%), and intraperitoneal type (8%); and the intraperitoneal type is the least Although pathologically benign, it is classified as intermediate/locally aggressive and non-metastasizing in the new 2013 World Health Organization tumor classification because of its characteristics of invasive growth, local recurrence, and no distant metastasis. We report a case of MDF ruptured to cause peritonitis, and resection of the tumor was possible after conservative treatment, without emergency surgery.This case report has been reported in accordance with the SCARE Criteria 2The patient was a 57-year-old man. He was emergently admitted to our hospital for lower abdominal pain and distension. There was no notable medical history, such as abdominal surgery, or family history. On admission, the entire abdomen was rigid, and muscle guarding was present, with the most significant tenderness in the lower right abdomen. Blood test findings revealed severe inflammation with a white blood cell count of 1000/μL, C-reactive protein level of 7.25 mg/dL and procalcitonin level of 8.6 ng/mL. Computed tomography (CT) revealed a 15 × 15 × 14-cm large internal heterogeneous intra-abdominal tumor with a mosaic-like contrast effect in the pelvis and the lower right abdomen, and abscess formation due to tumor necrosis was present on the ventral surface layer A. In addTherefore, we diagnosed acute peritonitis due to rupture following intra-abdominal tumor necrosis and intra-abdominal perforation. Since the patient's systematic symptoms were relatively stable, gastrointestinal perforation was ruled out, the differential diagnosis of the tumor itself was difficult at this point, and it was unclear whether or not the tumor could be resected even if emergency surgery was performed, we decided to start with conservative treatment such as fasting and antibacterial administration, and then proceed to differential diagnosis of the tumor, followed by treatment including surgery. When Meropenem 3 g/day was administered after hospital admission, the symptoms of peritonitis were improved the next day. Magnetic resonance imaging (MRI) examination revealed that the tumor had an internally non-uniform low signal on both T1- and T2-enhanced images, and fluid component retention was observed in the ruptured abscess A-B. The Subsequently, there was no intestinal obstruction due to water intake; however, CT re-examination revealed a still dilated small intestine, and there was a concern regarding resuming meals; therefore, laparotomy was performed on the 8th day after hospital admission.Intraoperative findings revealed that the tumor was located in the center of the ileocecal mesentery, and that the terminal ileum was involved in the tumor . HoweverThe specimen was a 165 × 125-mm large soft elastic tumor with a necrotic rupture site on the ventral side A. The suThe postoperative course was uneventful, and he was discharged on the 8th day after surgery (on the 16th day after admission). Since postoperative total colonoscopy found no polyposis, he was denied the diagnosis of familial adenomatous polyposis (FAP). Since complete resection was performed, postoperative adjuvant chemotherapy was not administered. There was no obvious recurrence at 1 year postoperatively.3DF was first reported by MacFarlane in 1833 The mechanism of tumor development is suggested to be associated with 10 to 20% of cases of hereditary diseases, such as FAP and Gardner's syndrome; mechanical stimuli, such as surgery and abdominal trauma; and a history of exposure to radiation appear to be involved as pathogenetic factors in sporadic development MDF exhibits few symptoms and is often found after becoming aware of an abdominal mass following tumor growth. However, cases of intestinal obstruction due to tumor invasion, and as in our case, peritonitis after tumor rupture have also been reported CT and MRI are frequently performed as diagnostic imaging. On MRI, DF appears as a wide low-intensity region on T1- and T2-weighted images and is often seen to have a non-uniform inside; however, sometimes it can present as a high-intensity region on T2-weighted imaging Surgical resection is the first-line treatment for MDF, but it has been reported that MDF recurred in 90% of patients with FAP and 11% of patients without FAP even after complete resection Although there were two cases in which the MDF itself ruptured to cause peritonitis, emergency surgery was performed in both cases 4MDF is often treated with surgery after thorough examinations. There are few reports of peritonitis caused by MDF rupture; emergency surgery can be avoided. Although complete resection of the tumor was possible in our case, local recurrence may occur in the future due to tumor rupture of the abdominal cavity, so careful follow-up is essential.This report has not received any funding.This is a case report and it did not require ethical approval from ethics committee according to our institution.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.Conception and design of the study: MT.Performed the surgery and perioperative management on the patient: MT, YM, HT.Data acquisition: MT.Drafting and revising of the article: MT, YM, TS, HT.Final approval of the version to be submitted: MT, YM, TS, HT.NA.Masahiro Tawada, MD.Not commissioned, externally peer-reviewed.The authors report no conflicts of interest.