Scrotal and retroperitoneal lymphangioma (SRL) in children is relatively rare and its clinical symptoms are usually difficult to distinguish from other conditions such as hydrocele and incarcerated inguinal oblique hernia. This study aimed to explore the clinical diagnosis and treatment of abdominal scrotal lymphangioma in children, and thus, to increase our understandings of this disease in clinical practice.This study enrolled nine boys, aged 1–10, who were admitted to Shanghai Children’s Hospital from January 2019 to December 2020 and who were finally confirmed with lymphangioma in the inguinal area. The clinical manifestations, diagnosis, and treatment of these children were analyzed retrospectively. The length of diagnostic process ranged from 3 weeks to 20 months. We also reviewed other cases of initially misdiagnosed cases of SRL in English publications from 2000 to 2022.The nine cases were misdiagnosed as hydrocele, hematoma, or inguinal hernia. Three patients received intracystic injection of bleomycin, three underwent laparoscopic mass resection, and three underwent resection of the inguinal lymphangioma under direct vision. Postoperative pathological analysis of the surgical specimens confirmed the diagnosis of benign cystic lesions and lymphangioma. Meanwhile, among the 14 cases of SRL in literature review, eight were misdiagnosed. Six were initially diagnosed as hydrocele, one as inguinal oblique hernia, and one as testicular tumor, all of which underwent ultrasonography scans. All cases were confirmed as lymphangioma after pathological examination.The non-specific clinical manifestations may contribute to the misdiagnosis of scrotal masses in children. A detailed and accurate medical history, careful physical examination, and imaging findings are important factors contributing to the preoperative differential diagnosis of scrotal lumps in children, but the final diagnosis is based on pathological examination. Lymphangiomas are benign tumors originating from the mesoderm. Their formation is characterized by abnormal development of the lymphatic system and they may occur in the head and neck (75%), axilla (20%), retroperitoneum, scrotum, and inguinal area (5%) . AlthougSRL is an extremely rare cause of scrotal lumps, which adds difficulties for practitioners to distinguish between SRL and other causes of scrotal masses, such as hydrocele, incarcerated inguinal hernia, and inguinal hematoma. Over the past 2 years, nine patients have sought medical advice in our center because of masses in the inguinal area. Of these, seven were initially misdiagnosed with hydrocele, one with incarcerated inguinal hernia, and one with inguinal hematoma, but all were proved to have lymphangioma by pathological staining after surgery.This retrospective study aimed to summarize the clinical manifestations and pathological results of these nine patients, and to analyze the reasons for their initial misdiagnoses.This study was approved by the Institutional Review Board of Shanghai Children’s Hospital, Shanghai Jiao Tong University, in accordance with the principles of the Declaration of Helsinki.This research reviewed the cases of nine boys aged 1–10 years who presented at Shanghai Children’s Hospital with unilateral inguinal masses and who were eventually diagnosed with SRL from January 2019 to December 2020. The time from onset to final diagnosis ranges from 3 weeks to 20 months. Among these nine cases, seven were initially misdiagnosed as hydrocele, one as inguinal hematoma with a clear history of trauma, and one as incarcerated inguinal oblique hernia  who were misdiagnosed with hydrocele presented with a painless mass in the scrotum and inguinal area, which grew slowly and proved to be cystic, without tenderness on physical examination. The masses measured 4 × 3 × 4 cm, 4 × 3 × 2 cm, and 3 × 2 × 2 cm, respectively. Transillumination was positive and the masses could be squeezed and narrowed.One child (Case 8) who was misdiagnosed with an incarcerated inguinal hernia displayed a red, swollen, and painful mass measuring 4 × 5 × 4 cm in the left inguinal region before surgery, having a hard texture and tenderness. The testis was slightly enlarged. However, the child vomited and refused to be touched and refused to cooperate with the transillumination examination Fig. .One child (Case 9) who was misdiagnosed with a left inguinal hematoma had a clear history of trauma in the left inguinal area 1 week before surgery. He complained of pain in the left inguinal area, and we detected a tender 3 × 4 × 4 cm soft mass without redness. The transillumination results were positive Fig. .The other four cases misdiagnosed with hydrocele  were diagnosed as lymphangioma in the inguinal area before surgery.Cases 1, 3, and 7 underwent ultrasound examination of the inguinal area within 3 months before surgery, which suggested hydrocele.9/L; hemoglobin, 88 g/L; and neutrophil percentage, 57.6%.One patient (Case 8) who was misdiagnosed with an incarcerated inguinal hernia appeared to show an inflammatory status and their brief complete blood count results were: white blood cells, 17.08 × 10Preoperative ultrasound imaging of Case 9 who was misdiagnosed with a left inguinal hematoma indicated a mixed mass in the left inguinal area, with the possibility of an inflammatory hematoma. The child also underwent preoperative contrast-enhanced computed tomography (CT), which showed mixed-intensity occupation, effusion, and bleeding Fig. .The remaining four cases  underwent ultrasound and pelvic CT and one (Case 4) also underwent magnetic resonance imaging, all of which suggested the possibility of lymphangioma.Seven children  underwent exploratory laparotomy. Three patients with huge lymphangiomas originating from the retroperitoneum received intrasaccular injection of bleomycin with no allergic adverse reactions, and the other four underwent laparoscopic mass resection. Case 1 underwent laparoscopic radical resection of the mass after 2 months. Case 7, who had undergone left laparoscopic high ligation of the processus vaginalis 2 years previously, showed severely adhered tissue around the cyst during laparoscopic exploration 2 months later. In addition, the left spermatic cord blood vessel surrounded the cyst and the posterior wall adhered to the left iliac vessels. Considering the risks associated with cyst resection, bleomycin was smeared onto the cyst in this patient. In Case 2, a large, dark red tumor with high tension on the left retroperitoneum extended into the left scrotum during surgery and dark red, non-coagulated blood was found by puncture. In Case 4, laparoscopic separation was difficult and a transverse incision was made in the right scrotum, allowing complete removal of the mass after separation. In Case 5, the intra-abdominal cyst was separated and removed via a dermatoglyphic incision in the left inguinal area by laparoscope-assisted surgery. The vas deferens adhered tightly to parts of the cyst walls and bleomycin was therefore applied. A follow-up ultrasound examination of the inguinal area 1 month later showed no signs of cystic lymphangioma expansion  underwent lymphangioma resection in the inguinal area under direct vision. In Case 9, the procedure was switched to inspection of the inguinal area under direct vision during surgery, which revealed an irregular bruising cyst closely linked to the spermatic cord and cremaster muscle. Considering a diagnosis of lymphangioma with hemorrhage, the tumor was resected completely.Postoperative pathological tests of the surgical specimens showed benign cystic lesions attributed to lymphangioma in all patients.The six children who underwent mass resection all had favorable prognoses at follow-up. In the three cases who received intrasaccular injection of bleomycin, the sizes of the tumors were obviously reduced and the patients were examined regularly by ultrasound every 2 months.We also identified 14 cases of SRLs reported in the English literature from 2000 to 2022 , ultrasound suggested a cystic mass and misdiagnosed it as a hydrocele. In addition, if ultrasound fails to detect if the mass extends into the abdominal cavity at the inner ring orifice, or if abdominal ultrasound is not performed, it can also lead to misdiagnosis of lymphangiomas. In some cases, such as Case 3 and in some emergency cases, preoperative ultrasound cannot be completed to determine the presence of incarceration of the intestines or other features such as time constraints, leading to a misdiagnosis of incarcerated inguinal hernia.Lymphangioma originates from the mesoderm and represents a congenital malformation of vascular development. It is not a ‘real’ tumor, but rather a benign lesion composed of multiple expanded lymphatic cysts formed by the abnormal development of the lymphatic system during the embryonic period. Lymphangiomas are more common in children, with about 90% diagnosed before the age of 2 years old. The incidences are equal between males and females , 15. ThePainless scrotal masses are usually benign, but a few are malignant, such as sarcomas . ScrotalVarious treatments are available for abdominal scrotal lymphangiomas. Surgical treatment with an inguinal approach is usually the first consideration –26. DuriPediatric abdominal scrotal lymphangioma is relatively rare and its clinical symptoms are often difficult to distinguish from those of other conditions such as hydrocele and incarcerated inguinal hernia. The diagnosis therefore depends mainly on a detailed and accurate medical history, careful physical examination, and imaging and pathological tests. Information on peritoneal scrotal lymphangioma is currently lacking. Unilocular peritoneal scrotal lymphangiomas can be resected directly and completely under laparoscopy, while surgical resection of multilocular lymphangiomas may damage the surrounding tissues, such as the vas deferens. In these case, sclerotherapy may be a safe and effective method, resulting in a favorable prognosis.This study was limited by the small number of cases, which meant that we were unable to verify the conclusions. In the future, we aim to compare the generalized patterns with clinical cases and further validate their practical value.SRL in children is a rare disease and its non-specific clinical manifestations lead to a high risk of its misdiagnosis among patients with scrotum masses as non-SRL conditions, including hydrocele and incarcerated inguinal oblique hernia. It is therefore necessary to obtain a detailed and accurate medical history and carry out careful physical examination, imaging, surgery, and pathological tests to make an accurate diagnosis. Unilocular peritoneal scrotal lymphangioma can be resected directly and completely under laparoscopy, while sclerotherapy is a safe and effective method for patients with multilocular lymphangioma.


Necrotizing enterocolitis (NEC) following postoperative stress is a rare but life-threatening condition in infants. We report a 3-month-old infant who underwent bilateral inguinal hernia repair and developed NEC. This is the first reported case of an infant developing recurrent NEC with stricture formation after herniotomy. Timely recognition and management are vital due to potentially high mortality rates in severe cases. High index of suspicion is crucial for accurate diagnosis and appropriate management. Necrotizing enterocolitis (NEC) is the most common life-threatening emergency affecting neonates . It is dA 3-month-old male infant was admitted to our pediatric department due to poor feeding and non-bilious vomiting for 3 days. He was born healthy at 36+2 weeks of gestation via C-section. The mother had COVID-19 infection twice during pregnancy and was successfully treated and recovered with no complications. His birth weight was 2.7 kg. The infant had been breastfed mainly with supplemental three times daily formula feeds. Family history was non-contributory.At 10 days of age, he started to have loud bowel sounds, projectile vomiting, and irritability, and he rejected the formula feeds only. Cow's milk protein allergy (CMPA) and gastro-esophageal reflux (GERD) were suspected. He was started on CMP-free formula and esomeprazole elsewhere. No improvement was noticed.At 20 days of age, he had an open bilateral inguinal herniotomy at another facility. After a few hours of the procedure, he developed rectal bleeding and abdominal distention. A gastrointestinal pathogens panel revealed norovirus and cryptosporidium. An abdominal ultrasound revealed portal vein gas but otherwise unremarkable findings. Bacterial translocation and NEC were suspected. Conservative treatment was initiated, and the infant relatively improved. Thus, he was discharged home. A few days later, the abdominal distention became worse, and the left inguinal hernia recurred. He had decreased feeding, recurrent episodes of whitish projectile vomit, infrequent loose stool, and poor weight gain. He was admitted six times to other facilities and then referred to our center.At the time of admission, the infant was lethargic and dehydrated. Abdominal distention, increased abdominal sounds, mild generalized tenderness, and a partially reducible left inguinal hernia were noted on the physical examination. Blood tests showed raised platelets, monocytes, C-reactive protein, procalcitonin, and normocytic normochromic anemia as illustrated in Table Contrast enema revealed significant narrowing of the right-middle transverse colon in keeping with stricture as illustrated in Figure The infant was taken to the operating room where rectal suction biopsies were performed and sent for a frozen section, followed by laparoscopic repair of the left recurrent inguinal hernia and a mini-laparotomy resection of the right-sided colonic stricture (6 cm length) with colo-colic anastomosis through a trans-umbilical incision. The frozen section showed a ganglionic bowel. The pathology of the resected right colonic stenosis showed segmental bowel wall fibrosis, loss of muscularis consistent with prior perforation, ulcerated mucosa with acute inflammation, serosal fibrovascular adhesions, and multiple benign reactive lymph nodes. The baby had an uneventful postoperative recovery and was discharged home after one week. He remains well 7 months after the procedure. Early (A), and late (B) postoperative appearance of the abdomen is shown in Figure Only a few cases of NEC due to postoperative noncardiac procedure-related stress have been reported in the literature. Comprehensive research was conducted in PubMed, Scopus, ScienceDirect, Embase, Web of Science, and Google Scholar from 1980 to date. Our review of the literature showed only 72 reported patients who had NEC after noncardiac surgeries, including myelomeningocele, intestinal atresia, gastroschisis, omphalocele, and congenital diaphragmatic hernia -10. The Our patient's clinical and radiological features were suggestive of NEC both before and after the first surgery. However, the infant improved with supportive therapy. We propose that the bowel was prone to inflammation, necrosis, and stricture formation due to formula feeding, previous NEC, and surgical stress. Amoury et al. related NEC to surgical stress caused by hypoperfusion resulting from increments in intraluminal pressure that lead to mucosal injury . In our All the previous papers highlighted abdominal distension, vomiting, and bleeding per rectum as demonstrations of NEC in babies at a median of 3 days postoperatively, all of which were encountered in our setting. The distinctive radiologic signs of NEC are pneumatosis intestinalis , gas in the portal vein, pneumoperitoneum, and dilated loops of the bowel ,2,4,9. NOverall mortality might reach 50% . For infFood protein-induced enterocolitis syndrome (FPIES) is a non-IgE-mediated allergic reaction typically triggered by cow's milk and soy protein. In neonates, it manifests as persistent emesis within 1-3 hours after feeding, accompanied by other symptoms such as lethargy, diarrhea, bloody stool, abdominal distension, anemia, eosinophilia, metabolic acidosis, and failure to thrive ,13. NotaThe combined laparoscopic and mini-laparotomy approach in our case led to ease in repairing the recurrent hernia by accessing it from a virgin area, and to optimal cosmetic results given the complex, multiple procedures he had concurrently. The resulting scars are almost invisible. We recommend that recurrent hernias are approached laparoscopically whenever possible even in the context of the need for simultaneous open surgery. The trans-umbilical incision is quite versatile in infants and can be used even for extensive bowel resection with excellent results .Though recurrence of postoperative NEC has been reported ,5,9, thiThis case highlights the importance of considering postoperative stress as a causative factor for complicated NEC, even when infants presents with the clinical and radiological manifestations of NEC after simple procedures such as inguinal hernia repair. Healthcare providers should have high index of suspicion to avoid devastating consequences.


The low sensitivity of ascites culture for acid‐fast bacilli necessitates a peritoneal biopsy when tuberculous peritonitis is suspected. Findings in the peritoneum on computed tomography may prompt suspicion of tuberculous peritonitis.A 47‐year‐old Nigerian man presented with fever, abdominal distention, and weight loss. Abdominal computed tomography revealed massive ascites and peritoneal thickening. Despite failing to culture acid‐fast bacilli from ascites, histological examination and culture of peritoneum revealed multidrug‐resistant tuberculosis peritonitis. Peritoneal biopsy is mandatory when tuberculosis peritonitis is suspected. Findings of abdominal contrast‐enhanced computed tomography in patients with tuberculous peritonitis. Mycobacterium tuberculosis. Laparoscopy showed white miliary nodules and fibrous adhesions  and transferred to a hospital that specializes in the treatment of tuberculosis.A 47‐year‐old Nigerian man who had been living in Japan for 20 years and had visited to Nigeria 3 years previously presented with lost 15 kg of body weight accompanied by abdominal distention without tenderness over 2 months. He had had a fever of over 38°C and watery diarrhea for 1 month. On admission, abdominal contrast‐enhanced computed tomography showed massive ascites, homogeneously thickened peritoneum, and enlarged mesenteric lymph nodes .Written informed consent was obtained from the patient to publish this report in accordance with the journal's patient consent policy.


Myxofibrosarcoma is a malignant mesenchymal tumor and a fibroblastic sarcoma of the elderly. Myxofibrosarcoma can be low-grade or high-grade depending on the cell characteristics. Wide surgical resection with or without radiotherapy and chemotherapy is the basis of its treatment. Sometimes, tumor cells secrete insulin or insulin-like substances and cause hypoglycemia attacks. Here, we intend to demonstrate the role of early surgery to end hypoglycemia attacks and prevent recurrence and metastases. We also intend to show the insufficiency of tru-cut biopsy to distinguish between low- and high-grade myxofibrosarcoma. An 82-year-old male patient visited our clinic with a rapidly growing giant mass in the left retroscapular area and suffered from hypoglycemic attacks several times a day. After imaging and initial biopsy, the tumor grade was indeterminate on histopathological examination; hence, the mass was removed surgically. The pathological examination resulted in high-grade myxofibrosarcoma whereas the initial biopsy could not elaborate on the grade. The hypoglycemia attacks ceased after the surgery. Adjuvant local radiotherapy at a total dose of 60 Gy was administered in 30 fractions to the surgery area with no complications after the surgery. No new mass, recurrence, or hypoglycemia attack was detected in the three-year follow-up. In conclusion, hypoglycemia attacks may be a marker of malignant tumor presence and may be a clue at the beginning and in the follow-up period both for recurrence and the aggressiveness of the tumoral mass. Because a biopsy may show the diagnosis but not the grade of the tumor, early surgical intervention is needed. Although myxofibrosarcoma, known as myxoid malignant fibrous histiocytoma, is a rare soft tissue spindle cell malignant mesenchymal tumor, it is the most common fibroblastic sarcoma in older patients . MyxofibAn 82-year-old male patient visited our clinic with a painful mass presenting a high growth tendency in the left retroscapular area. The mass first appeared five months ago and showed rapid growth in the past two months Figure .The patient suffered from hypoglycemic attacks several times a day as his glucose level ranged between 45 and 55 mg/dL. The patient’s potassium levels also ranged between 2.5 and 3.5 mEq/L, indicating mild hypokalemia during the attacks. A space-occupying lesion was detected on the posteroanterior and lateral chest radiograph   with the capsule. The excised material was sent to pathology for examination  because early surgical excision was performed without local and systemic invasion of the tumor. In this way, the patient’s survival and quality of life were improved. Because the tumor was high grade and larger than 5 cm, the patient was subjected to frequent clinical follow-ups every three months in the first year, every six months in the second year, and once a year following that. In addition, the result of a tru-cut biopsy is insufficient to distinguish between low-, medium-, and high-grade myxofibrosarcoma and the importance of clean surgical margins and en bloc resection. Moreover, early surgical intervention will enable surgeons to prevent hypoglycemic attacks after surgical removal of the total mass so that the patient’s internal medicine problems can be resolved without medication. The effect of IGF signaling and IGF-2 redundancy on paraneoplastic hypoglycemia should be a focus of targeted therapy in further investigations.For the diagnosis of myxofibrosarcoma, the biopsy may show myofibroblastic differentiation but not the grade of the tumor. Therefore, early surgical intervention with wide resection is needed as it resulted in high-grade myxofibrosarcoma in our case. Hypoglycemia attacks may be a sign of the presence of a malignant tumor and may provide information about recurrence and the aggressiveness of the tumoral mass both at the beginning and during the follow-up period. In future research, the radioimmunoassay approach, which was lacking in our case study, may be used to identify the paraneoplastic-released chemical triggering hypoglycemic crises.


Recurrent liposarcoma, previously confirmed as lipoma, has rarely been reported. However, the risk factors for recurrence and the correlation between benign lipoma and malignant liposarcoma remain unclear. In this case study, we suggest a precise diagnostic strategy to minimize recurrence and malignant transformation.A 60-year-old male patient with a history of left chest wall swelling without any symptoms underwent excisional surgery, and the mass was confirmed as a benign lipoma in 2015. In 2019, the patient returned to the hospital with symptoms of a palpable mass on the left chest wall.The mass was considered a recurrent lipomatous tumor with the possibility of malignant transformation. Magnetic resonance imaging (MRI) revealed a deep-seated, septate, intramuscular, irregular margin, and large lipomatous tumor invading the ribs, pleura, and adjacent muscle, suggestive of malignancy. The MRI findings were similar to those 4 years ago, except for margin irregularity and invasion to adjacent tissue.Wide en bloc excisions encompassing the 5th to 7th ribs, pleura, and adjacent muscle were followed by reconstruction with a pedicled latissimus dorsi muscle flap.The recurrent large lipomatous tumor was confirmed as well-differentiated liposarcomas through histological and MDM2-FISH immunohistochemical staining. Postoperatively, follow-up visits continued for 1.5 years without recurrence.We suggest that deep-seated, septate, and giant lipomatous tumors should be considered as risk factors for recurrence with the possibility of malignancy and misdiagnosis. It is important to inform patients of all these possibilities and plan close and long-term follow-up. It is the most common sarcoma accounting for 10% to 15% of all cases  Moreover, it could be difficult to distinguish these 2 masses completely via intraoperative frozen section examination because of similar histological characteristics. Histologically, WDLS is characterized by mature fat with a variable number of spindled cells displaying hyperchromatic nuclei and multi-vacuolated lipoblasts, testing positive for CDK4 (average 62.1%) immunohistochemically. Most benign lipomas consist of mature adipocytes, but a few lipomas are similar to WDLS not only in histology but also in immunochemistry. making it difficult for many surgeons to distinguish between benign and malignant cases.The differential diagnosis of benign lipoma and WDLS is difficult based on preoperative imaging studies alone, especially if they are large in size, located in the intramuscular layer, or are septate with unclear boundaries. The incidence of recurrence is thought to be reduced by performing precise assessment considering the possibility of malignancy with the risk factors and understanding the correlation between benign lipoma and WDLS.Recurrence of lipomatous tumor as malignant liposarcoma after surgical excision of benign lipoma has rarely been reported. while other studies have reported the possible origin of these 2 masses from the same adipocytes with identical genomic background. On the other hand, there have been reports of co-existence of both masses.–12 However, its pathogenesis remains unclear. In this case study, we present a case of recurrent liposarcoma that was surgically excised and confirmed as a benign lipoma. In this case, we want to discuss the correlation between benign lipoma and WDLS along with a differential diagnosis, to present the risk factors of recurrence, and to consider ways to reduce recurrence with malignant transformation.In an effort to understand the pathogenetic correlation between 2 masses, several recent studies have raised the possibility of a biological continuum of benign, atypical, and malignant mesenchymal neoplasms and malignant transformation from benign lipoma to WDLS.22.1This study was approved by the Institutional Review Board of the Catholic Medical Center Office of the Human Research Protection Program. (KC20ZASA0156) at the Catholic University of Korea . All data were analyzed anonymously and according to the principles of the Declaration of Helsinki 1975 (revised in 2008).The patient has provided informed consent for publication of the case.3A 60-year-old man presented to the Department of Plastic and Reconstructive Surgery with a history of left chest wall swelling without any symptoms in 2015  and variation in cell size, along with focal adipocytic nuclear hyperchromasia Fig.  B. The h results of MDM2-FISH showed focally positive and 5.96 of MDM2/CEP12 ratio, suggesting that MDM2 gene amplification indicated the lipomatous tumor as WDLS.Immunochemical staining with MDM2-FISH was performed to examine gene amplification. MDM2 and chromosome 12 centromere (CEP12) using the Vysis LSI MDM2 Spectrum Orange Probe nd Vysis DNA FISH probe CEP12  were examined, and.The patient was discharged after postoperative care. Postoperatively, follow-up visits continued for 1.5 years. Follow-up CT for screening of recurrence was conducted a year postoperatively, and no evidence of recurrence was found. The patient remained stable without recurrence Fig.  B.4 In this study, a deep-seated, septate, and large-sized lipoma was detected on the MRI images in 2015. Although this tumor was considered to be a benign lipoma most likely due to low heterogeneity and regular margin findings, the features described above are thought to suggest a potential risk of recurrence. Therefore, considering these risk factors and performing precise preoperative evaluation is required to prevent the recurrence of lipoma with malignant transformation.A case study of recurrent WDLS on the forearm flexor compartment was reported in which a benign lipoma was surgically excised 28 years ago. According to this study, the risk factor for recurrence with malignancies is presented as a large-sized (giant) lipoma of 5 cm or more, and unusual characteristics in the location of the tumor. MRI images that suspect liposarcoma include size, depth, nodularity, or stranding as well as heterogeneity; recently, based on radiomic-MR imaging, texture analysis could contribute to the advancement of differential diagnosis by quantifying the texture heterogeneity of lipomatous tumors.,17In the present case of deep-seated, septate, and large-sized lipomatous tumors, differential diagnosis from liposarcoma before surgery could be difficult with unclear radiological findings, and the possibility of liposarcoma cannot be completely excluded before surgery. Therefore, surgeons must select appropriate radiological examinations and understand the radiological findings suggesting malignancy. Among the radiological examinations, magnetic resonance imaging (MRI) has been considered the gold standard for the differential diagnosis of soft-tissue tumors.However, performing MRI on all patients visiting with soft-tissue tumors is less cost-effective, so it is necessary to select an appropriate screening modality according to the characteristics of the tumor. Based on this appropriate screening modality, a patient-specific approach is needed for accurate decision-making. According to the recently presented uncontrolled retrospective study, in which the differential diagnosis between WDLS and benign liposarcoma was performed using sonography imaging, sonographic findings were reported to predict WDLS with highly accurate correlation with pathological features. The features of malignancy are described as1.deep location,2.irregular shape,3.hyperechogenicity,4.large size, and5.presence of vascularity.; therefore, ultrasonography could be considered as a beneficial screening modality for malignant lipomatous neoplasm with non-invasiveness and cost-effectiveness. We suggest that sonographic examination should first be conducted with a screening modality for patients complaining of lipomatous tumors, and perform more accurate MRI examinations for patients with malignant features on sonographic examination.In addition, and contrast-enhanced color Doppler ultrasonography could find the probability of malignancy with advanced accuracy, not only by enhancing the margin, depth, textural pattern of malignant soft tissue tumor on gray-scale US, and by visualizing vividly fine vascular structures developed in malignant tumors Although the possibility of co-existence, some histological sections with clustered and mosaic elements with and without cell atypia in the WDLS could mask the atypia and lead to a diagnosis of benign lipoma.After surgical removal of the lipomatous tumor, surgeons need to question the precision of the pathologic finding. In this case, a deep-seated, septate, intramuscular, and large-sized lipoma was pathologically confirmed as a benign lipoma after complete surgical excision in 2015. As reported in the previous literature, in the case of lipomatous tumors with this characteristic, the possibility of co-existence of both benign and malignant masses cannot be excluded. In this case, the patient was diagnosed with adipocytic WDLS. Differential diagnosis of adipocytic WDLS from large benign lipoma occasionally presents similar histological characteristics, whereby the WDLS was identified predominantly with mature fat containing variable numbers of spindled cells displaying hyperchromatic nuclei and multivacuolated lipoblasts, immunohistochemistry is needed to perform accurate pathologic diagnosis of liposarcomas from benign lipomas. Currently, MDM2 and CDK4 using FISH immunochemical staining are considered as criterion standard methods for IHC.–25 However, errors in interpretation of ICH have been reported in which benign lipomas with secondary changes with inflammatory cells stain positive with MDM2  To diminish the challenges of histological diagnosis, P16 immunohistochemistry as an alternative marker combined with the MDM2-FISH immunochemical staining have recently been suggested as a diagnostic method to distinguish WDLS from deep-seated lipoma.,26WDLSs can be classified as adipocytic, sclerosing, inflammatory, or spindle cells based on pathomorphology.As described above, in this patient, a large, deep-seated lipomatous mass, which was diagnosed as benign lipoma in 2015 and underwent surgical excision, recurred at the same site 4 years later as malignant liposarcoma with invasion to the ribs, pleural tissue, and surrounding muscles. In order to prevent recurrence that has progressed to malignancy, surgeons need to clarify the possibility of malignancy with appropriate imaging tests before surgery, to inform radiologists about the precise clinical impression that can lead to improved accuracy of assessment of malignant lipomatous tumors. If the radiological examination is unclear, in the case of lipomatous tumors showing a risk factor for recurrence before surgery, we suggest to perform surgical excision including an appropriate safety margin. For pathologic confirmation, if a malignant lipomatous tumor is strongly suspected before surgery, even if benign lipoma is confirmed by histological staining, we propose to perform immunohistochemistry considering the possibility of misdiagnosis on conditions agreed upon by the patient.5Through this case study, we suggest that deep-seated, septate, and giant lipomatous tumors should be considered as risk factors for recurrence with the possibility of malignancy and misdiagnosis. It is important to inform patients of all these possibilities and plan close and long-term follow-up.To prevent recurrence, it is necessary to surgically confirm the safety margin for excision, to request a careful pathological examination of all histological sections based on the focality of nuclear atypia.Data curation: Yeon Ji Lee, Won Jin Cha, Yesol Kim.Supervision: Deuk Young Oh.Validation: Deuk Young Oh.Visualization: Won Jin Cha, Yesol Kim.Writing – original draft: Yeon Ji Lee.Writing – review & editing: Deuk Young Oh.


Aurora rings are prominent endoscopic features of the inverted colonic diverticulum (ICD), and their appearance alongside a colonic lipoma is an unprecedented phenomenon. The present study reports a case of colonic lipoma with Aurora rings, contradicting the assumption that Aurora rings are indicative of ICD. A 52-year-old male patient presented with left-sided abdominal pain for >1 year, associated with constipation in the form of the decreased frequency of bowel motions every 4 to 5 days. A physical examination revealed an obese, protuberant abdomen and a mildly tender left iliac fossa region without other notable findings. A transabdominal ultrasonography revealed a thickening of the large bowel wall (<7 mm) with a suspected inflammatory lesion on the left side of the colon. During an ileocolonoscopy, multiple diffuse diverticula of various sizes were observed, affecting the entire colonic mucosa. Furthermore, a large (1.5 cm) pedunculated polyp with a thick stalk was found in the sigmoid colon, exhibiting positive Aurora rings. A polypectomy was conducted with the deployment of two hemoclips at the polyp base to prevent perforation. The histopathological examination of the specimen, a 1.3 cm polyp, revealed the presence of a colonic lipoma, rather than an ICD. The identification of Aurora rings has emerged as a significant endoscopic feature in the diagnosis of ICD; nevertheless, the underlying etiology of these rings remains elusive. Based on an extensive search of the literature, no study was found mentioning the appearance of Aurora rings in an endoscopic screening of other colonic conditions other than ICD. The appearance of Aurora rings alongside a colonic lipoma has not previously been mentioned, at least to the best of our knowledge, which renders the differentiation of ICD from lipomas and polyps more challenging. Diverticula refer to mucosal and submucosal pouches bulging from the muscularis propria. Inverted colonic diverticulum (ICD) is an infrequent incident occurring in ~0.7-1.7% of the general population, with a few reported cases in the literature . ColonicTo the best of our knowledge, the appearance of Aurora rings with a colonic lipoma in colonoscopy screening has not yet been mentioned. The present study reports a case of colonic lipoma with Aurora rings, which contradicts the assumption that Aurora rings are indicative or pathognomonic of ICD.A 52-year-old male patient presented to the GIT Department, at Smart Health Tower  with left-sided abdominal pain for >1 year, associated with constipation in the form of decreased bowel movements every 4 to 5 days. The patient had a good appetite and did not experience weight loss, rectal bleeding, or any notable previous surgical history. The only known medical condition was hypertension, for which the patient was taking hydrochlorothiazide. The patient was an outpatient who had ignored his condition until the pain progressed to severe pain, and he then visited the hospital for his condition.A physical examination revealed an obese, protuberant abdomen and a mildly tender left iliac fossa region. There were no other notable findings.Blood examinations revealed normal blood glucose and serum creatinine levels, and a complete blood count. A transabdominal ultrasonography (US) revealed a thickening of the large bowel wall (<7 mm), with a suspected inflammatory lesion on the left side of the colon. There was no obvious collection or mass. An ileocolonoscopy revealed multiple, different-sized diffuse diverticula involving the entire colonic mucosa, with a large (1.5 cm) thick stalk pedunculated polyp in the sigmoid colon . The polFollowing discussions with an expert gastrointestinal surgeon, a polypectomy through endoscopy was conducted with the deployment of two hemoclips at the polyp base to prevent perforation, as an ICD was suspected in this case. A histopathological examination was performed under the following conditions: The sections (4-5 µm-thick) were paraffin-embedded and fixed with 10% neutral-buffered formalin at room temperature for 24 h. The sections were then stained with hematoxylin and eosin (Bio Optica Co.) for 1-2 min at room temperature and examined under a light microscope (Leica Microsystems GmbH). The histopathological examination of a specimen  revealed the presence of a colonic lipoma, rather than an ICD .Post-operatively, the patient had only mild abdominal pain, and he was admitted to the hospital for 24 h. Later on, his condition recovered, and the patient was discharged.The ICD is a rare condition, affecting ~0.7-1.7% of the general population, with a higher prevalence among males. It predominantly occurs in the sigmoid colon and manifests as a polypoid lesion, presenting either as a sessile morphology or pedunculated structures . It is cRecently, scholars have focused on techniques that can identify the main features of an ICD, aiding endoscopists in making better diagnoses. The radiating pillow sign has been reported to be critical in differentiating an ICD from a lipoma ,8. FurthThese concentric rings were first described during the endoscopic screening of the colon in several cases by a gastrointestinal technical specialist named Aurora. Aurora and her colleagues claimed that visualizing these rings may provide evidence of ICD without the need for endoscopic maneuvers . After AColonic lipoma is a rare benign tumor with an unknown etiology that may be asymptomatic or cause blood loss, abdominal pain and bowel obstruction . This tuThe management of colonic lipoma depends on the lesion size and presentation. Small, asymptomatic lesions may only require follow-up. Symptomatic lesions or asymptomatic lesions >2 cm in size require resection, either surgical or endoscopic. Endoscopic resection is considered more suitable for managing symptomatic lipomas that are <2 cm in diameter or are pedunculated. However, it should be noted that performing endoscopic resection on lipomas >2 cm is associated with a greater risk of complications, such as hemorrhaging and perforation . In addiIn conclusion, the presence of Aurora rings with a colonic lipoma has not been previously reported, at least to the best of our knowledge. The finding of this report challenges the assumption that Aurora rings are solely indicative of ICD. Consequently, distinguishing ICD from lipomas and polyps becomes more complex, thus necessitating an extensive workup.


Muscle herniation of the lower extremity, such as tibialis anterior muscle herniation (TAMH), is not a rare cause of leg pain in athletes. However, a few studies have reported surgical treatment for TAMH, and the optimal surgical procedure remains controversial. Fasciotomy was reported to be effective for patients with TAMH. However, this procedure would be associated with a risk of intraoperative injury to the superficial peroneal nerve (SPN), although no previous literature has reported this complication. This case report aimed to report a case of bilateral TAMHs in which a traumatic neuroma of the SPN developed after fasciotomy. A 16-year-old baseball player presented with painful swelling lesions of the bilateral lower extremities  after sports activities. An ultrasonographic evaluation showed swelling lesions of the anterolateral parts of the bilateral lower extremities in the standing position after dashing, while these lesions were not detected in the supine position. A fasciotomy of the crural fascia was performed after conservative treatment failed. Several days after surgery, the patient presented with weakened touch sensation over the dorsal area of the left foot. At the three-month follow-up examination, a swelling lesion with hard elasticity was identified. The palpation of this lesion caused a radiating sensation in the area supplied by the SPN. He was able to return to playing baseball six months after surgery. The patient was asymptomatic without palpation of the traumatic neuroma of the SPN at the latest follow-up examination. In conclusion, the present case report suggests that orthopedic surgeons need to consider the risk of iatrogenic injury to the SPN during fasciotomy for the treatment of TAMHs. However, there may be a risk of injuring the SPN because of the many variants of the course of the SPN within the compartment of the lower extremities. Muscle herniation of the lower extremity is reported to occur after blunt traumatic injury to the lower leg or muscle hypertrophy due to strenuous activities in athletes or soldiers ,2. Lee eWhen fasciotomy is performed for the treatment of TAMH, similar to that for the treatment of chronic exertional compartment syndrome (CECS) of the lower leg, clinicians should pay attention to iatrogenic damage to the superficial peroneal nerve (SPN) ,10,11,12This case report aimed to show a case of bilateral multiple TAMHs in which a traumatic neuroma of the SPN developed following fasciotomy. Informed consent was obtained from the patient and his parents for the publication of this report and any accompanying images.A 16-year-old baseball player was referred to our hospital for the assessment of multiple swelling lesions in the bilateral anterolateral lower legs. He had suffered from painful lesions when playing baseball for about a year, limiting his athletic performance. He had no history of traumatic injuries to the lower legs. Clinical examinations showed bilateral painful swelling lesions (1 on the right and 3 on the left) after dashing . These lFasciotomy of the crural fascia was performed after two months of conservative treatment failed. The surgery was performed under spinal anesthesia with the patient in the supine position. A tourniquet was used for exsanguination. Firstly, a 30-mm longitudinal incision was made over the lesion in the right lower leg. When the crural fascia was exposed, thin fascia was detected A. A fascPostoperatively, the ankles were not immobilized, and weight bearing was allowed as tolerated. Ankle range-of-motion exercises were initiated immediately after surgery. Several days after surgery, he presented with paresthesia and impaired touch sensation over the dorsal area of the left foot. At the three-month follow-up, a swelling lesion with hard elasticity was identified between the middle and the most distal surgical wounds. The lesion did not disappear with the patient in the supine position. On palpating this lesion, he recognized a radiating sensation over the left dorsal foot supplied by the SPN. An ultrasonographic examination showed an isolated lesion (8.0 × 12.0 mm) . MRI shoFirst, we presented a case of bilateral multiple TAMHs with traumatic neuromas of the SPN following fasciotomy. Several surgical procedures have been reported for the treatment of TAMH ,6,7,8,15In the present case, traumatic neuroma of the SPN occurred after fasciotomy. Several studies have demonstrated that there are many variants of the course of the SPN and its branches within the compartment of the lower extremities ,14,16,17We presented a case of bilateral multiple TAMHs in which a traumatic neuroma of the SPN developed after fasciotomy. For the treatment of TAMHs, clinicians need to consider the risk of iatrogenic injury to the SPN during fasciotomy.


Sarcomas are a rare type of cancer that can develop in various parts of the body, including the brachial plexus. Leiomyosarcomas (LMs) are a subtype of sarcoma that develops in smooth muscle tissue and can metastasize to different parts of the body. In this case report, we present two patients with LM metastasized to the brachial plexus, one treated with CyberKnife  stereotactic radiosurgery (CK SRS) and the other with surgical resection. The aim of this case report is to present the treatment outcomes and adverse effects of CK SRS and surgical resection in brachial plexus LM metastasis. Patient 1 was a 39-year-old female who received CK SRS, and at three months of follow-up, the lesion was smaller, and she reported symptomatic improvement. At 15 months, the lesion was stable in size, and there was no evidence of local invasion of the adjacent vascular structures or nerves. Patient 2 was a 52-year-old male who underwent surgical resection, and at one-month follow-up, the patient was asymptomatic with no recurrence of his symptoms. The size of the residual axillary tumor was stable at three months and showed a slight interval decrease in size at five months of follow-up. He was followed for over 12 months, with no recurrence of his symptoms.Both treatments appear to have been effective in controlling LM growth and relieving symptoms. CK SRS provides a non-invasive option. However, more research is needed to fully understand the effectiveness and safety of these treatments for brachial plexus sarcoma. This case report highlights the importance of considering different treatment options for brachial plexus sarcoma and the need for further studies to understand the best approach for these rare cases. Sarcomas are a rare type of cancer that can develop in various parts of the body , includiThe management of LMs is multifaceted and requires a multidisciplinary team approach. Surgical intervention remains the cornerstone of treatment for localized disease, with radiation therapy employed as an adjunct modality to optimize local control. Chemotherapy is reserved for patients with metastatic or unresectable tumors and may be used as adjuvant therapy in selected cases . In thisMethodsThis manuscript is drafted using Chat Generative Pre-trained Transformer (ChatGPT), a large language model trained by OpenAI Figure . ChatGPTCase 1Patient 1 is a 39-year-old female with a history of retroperitoneal spindle cell LM that metastasized to the pancreas, lung, and spine. LM metastasized to the spine at T6 and T12 levels and was treated with CK SRS in combination with chemoradiation. A marginal dose of 24 Gy was delivered in three fractions to each lesion to treat a volume of 13.3 and 5.27 cc, respectively. The chemotherapy regimen used was gemcitabine-docetaxel, temozolomide, and sorafenib. At the age of 43 years, she had a progressing disease and presented with the symptoms of left arm numbness and occasional left shoulder pain. She also had pain and tingling in her thumb and forearm on the same side. Chest MRI demonstrated a contrast-enhancing mass on the left, adjacent to the brachial plexus and anterior to the subclavian vein measuring 1.6 cm x 1.0 cm x 1.0 cm , and brachial plexus. The metastatic lesions were discovered at the age of 58 years when he presented with generalized weakness and shortness of breath. He presented with weakness, dyspnea, and presyncope and was found to have a mass on imaging. Chest CT demonstrated a right hilar mass extending into the left atrium and right pulmonary vein along with a right axillary mass. A shoulder MRI showed a T1 isointense and T2 heterogeneously hyperintense enhancing mass with central hypo-enhancement in the right axilla , and she reported symptomatic improvement. At seven months, the lesion was stable in size, and there was no evidence of local invasion of the adjacent vascular structures or nerves. The patient had no left arm numbness or shoulder pain anymore. The most recent MRI was performed at 15 months, which showed no evidence of local progression. The patient remained asymptomatic for the brachial plexus metastasis. Progression-free survival (PFS) was 18.6 months. The patient died at this time due to aggravation of the primary disease. During follow-up, the patient did not experience any radiation adverse effects.Patient 2 underwent a complicated surgery, with a pneumothorax needing temporary chest tube placement. At the one-month follow-up, the patient was asymptomatic with no evidence of recurrent pneumothorax. The size of the residual axillary tumor was stable at three months and showed a slight interval decrease in size to 2.4 cm x 2.1 cm x 1.6 cm at five months of follow-up (Figure Both cases presented in this report have in common the presence of LM metastasized to the brachial plexus. However, there are differences in terms of the treatment received, with patient 1 being treated with CK SRS and patient 2 undergoing surgical resection. Similar cases of brachial plexus sarcomas have been previously reported in the literature -7, with The general management of leiomyosarcoma involves a multidisciplinary approach. Surgery is the primary treatment for localized disease, and the goal is to achieve negative margins. Radiation therapy may be used in addition to surgery to improve local control. Chemotherapy is typically reserved for metastatic or unresectable diseases and may be used as adjuvant therapy in certain cases. In cases where the tumor is unresectable, palliative care is the mainstay of treatment. Close monitoring and surveillance are crucial for detecting recurrence or metastasis. The management of leiomyosarcoma requires individualized treatment plans tailored to the patient's specific needs and disease characteristics .Stereotactic radiosurgery, such as CK SRS, is a non-invasive method that uses high-dose radiation to target and destroy cancer cells. The available evidence for its effectiveness in treating leiomyosarcomas is limited, with some studies showing promising results in terms of local control and symptom relief. However, more research is needed to fully understand the effectiveness of CK SRS in treating LM metastases to the brachial plexus.In terms of surgical resection, it is considered a standard of care for most sarcomas . SurgeryThe cases presented in this report suggest that both CK SRS and surgical resection can be effective in treating LM metastases to the brachial plexus, but CK SRS may have an advantage in terms of avoiding surgical complications . HoweverThis case report presents two cases of LM metastasized to the brachial plexus, one treated with CK SRS and the other with surgical resection. Both treatments appear to have been effective in controlling the LM growth and relieving symptoms, with CK SRS providing a non-invasive option and surgical resection providing good local control. However, more research is needed to fully understand the effectiveness and safety of these treatments for brachial plexus sarcoma. The take-home message is that brachial plexus sarcoma is a rare disease, and more research is needed to understand the best treatment options.


Eccrine poroma (EP) is a benign adnexal tumor that is derived from acrosyringium, the intraepidermal eccrine duct of sweat glands. The standard treatment for eccrine poroma is complete excision. However, this case report highlights cryotherapy as one of the modalities in treating eccrine poroma. We present a case of a 33-year-old male patient who was a known case of generalized vitiligo since he was nine years old. During our skin checkup before starting him on phototherapy, we found a mass over the palmar aspect of the middle finger of the right hand that started to appear five years ago. The mass gradually increased in size, was painless, has no discharge, and was not associated with a history of trauma or infection. The review of systems was unremarkable. Skin examination revealed an asymptomatic, 2.0 × 1.5 cm-sized, solitary, collarette-encircled, dome-shaped, flesh-colored, non-pigmented, deep-red nodule protrusion from the palmar aspect of the middle finger of the right hand. Poroma was considered as the diagnosis, and a punch skin biopsy was performed to confirm the diagnosis and to roll out pyogenic granuloma, amelanotic melanoma, and porocarcinoma as differential diagnoses. A 3 mm punch skin biopsy was performed under local anesthesia and was found to be histologically consistent with eccrine poroma. Hence, cryosurgery was chosen based on histological favorable features. We used cryospray in a single session of 15 seconds in three applications, with five-second intervals in between (skin frosting recovery). Furthermore, the lesion was completely curative with a single session of cryotherapy. The patient followed up for one year without evidence of recurrence. Eccrine poroma (EP) is a benign adnexal tumor that is derived from the acrosyringium, the intraepidermal eccrine duct of sweat glands . Sweat gA 33-year-old male patient, a known case of generalized vitiligo since he was nine years old, was recently started on narrowband ultraviolet B (NB UVB) and then shifted to excimer laser because the patient could not tolerate excessive heat. During the skin checkup before the beginning of his phototherapy, we found a mass over the palmar aspect of the middle finger of the right hand that started to appear five years ago. The mass gradually increased in size, was painless, has no discharge, and was not associated with a history of trauma or infection. The review of systems was unremarkable. Examination revealed an asymptomatic, 2.0 × 1.5 cm-sized, solitary, collarette-encircled, dome-shaped, flesh-colored, non-pigmented, deep-red nodule protrusion from the palmar aspect of the middle finger of the right hand  Figure .Furthermore, the lesion was completely curative with a single session of cryotherapy. Then, the patient followed up for one year without evidence of recurrence Figure .Our case study is unique in the treatment modality since the lesion was resolved after a single session of cryotherapy and followed up for one year without evidence of recurrence. The standard for EP is complete excision to prevent recurrence . On the For the treatment of benign and solitary eccrine poroma, cryotherapy is an efficient and well-tolerated method whose advantages include quick execution, lack of need for local anesthesia, and low cost. However, there are no established guidelines for when cryotherapy should be applied to various parts of the body and for how long to freeze and thaw. In this context, randomized controlled clinical trials (RCTs) are required to improve the procedure and reduce the risk of over- or under-treatment.


Myelolipoma is a benign neoplasm of the adrenal cortex, composed of fat and hematopoietic cells. Although myelolipoma is benign, differentiation from adrenocortical cancer may be difficult. The presence of adrenal and extra-adrenal myelolipomas simultaneously is sporadic, making it a challenging case, especially when the preoperative diagnosis is ambiguous.A 65-year-old man was referred to our clinic due to a mass in the adrenal fossa. In the abdominopelvic computed tomography (CT), a well-circumscribed fat-containing 78 × 61 × 65 mm bi-lobulated mass was reported in the left adrenal fossa. The first differential diagnosis was myelolipoma. The patient was then referred to our clinic for a mass excision. He was asymptomatic and was scheduled to undergo laparoscopic-assisted adrenalectomy. After adrenalectomy and mass dissection, surprisingly, another mass was detected in the retroperitoneal area. The second mass was also dissected. The final diagnosis was myelolipoma for both masses. The patient has been symptom-free for nine months after the operation.Simultaneous adrenal and extra-adrenal myelolipoma should be considered as one of the differential diagnoses. However, because this situation is extremely rare, the probability of malignancy should be highly regarded, and we suggest an obsessive approach when approaching this condition. It is essential to manage these cases on a case-by-case basis and tailor the management concerning intraoperative biopsy, the intraoperative appearance of tumors, and the location of extra-adrenal masses. Myelolipoma is a benign neoplasm of the adrenal cortex composed of fat and hematopoietic cells , 2. MostMyelolipomatous lesions have three patterns on imaging; isolated adrenal myelolipoma, myelolipoma with hemorrhage, and myelolipomatous foci with other adrenal pathologic conditions , 7. Alth2, the vital signs were stable, and except for the absence of the thyroid gland, the physical exam did not show any abnormalities.A 65-year-old man with a past medical history of papillary thyroid carcinoma and eye cataract was referred to our clinic due to a mass in the adrenal fossa. On admission, the patient had an approximate body mass index (BMI) of 30 kg/mThe patient underwent radical thyroidectomy due to papillary thyroid carcinoma and took levothyroxine. He had been under follow-up with an endocrinologist regarding this issue. On one of his visits to the endocrinologist, calcium, phosphorous, and hormonal assessment alongside neck, chest and mediastinum, and abdominopelvic computed tomography (CT) scan was ordered to be performed. In the blood test, calcium, phosphorous, T4, antithyroglobulin antibody, and thyroglobulin were within the normal range, but thyroid-stimulating hormone (TSH) was 0.05 (reference range: 0.27–5.95). Also, 24-hour urine levels of cortisol, vanillylmandelic acid (VMA), metanephrine, and normetanephrine, measured to exclude the presence of a possible pheochromocytoma, were within the normal range. However, in the abdominopelvic CT, a well-circumscribed fat-containing 78 × 61 × 65 mm bi-lobulated mass was detected in the left adrenal fossa, resulting in inferior and posterior displacement of the left kidney. The first differential diagnosis was myelolipoma. Besides, no enhancing nodule or solid part was seen .In the literature, simultaneous adrenal and extra-adrenal myelolipomas are reported exceptionally rarely. The importance of such cases is their challenging management due to the rarity of these situations and the ambiguity of pathologies in the pre-surgical diagnosis. A case report by Zieker et al.  presenteThe clinical diagnosis of myelolipomas is impractical because most myelolipomas are asymptomatic and are found incidentally. Based on the study by Hamidi et al. , which eImaging tools and biopsy are helpful tools for the diagnosis of myelolipomas. In ultrasonography, as the primary imaging tool, myelolipomas have variable appearances based on their composition. Although ultrasound is limited for diagnosis, especially when the mass is small, myelolipomas typically appear as a heterogeneous hyperechoic mass , 16. HowThe differential diagnoses of fat-containing retroperitoneal masses include tumors originating from adrenal , kidney (angiomyolipoma), and pancreas , and primary retroperitoneal masses  . Of noteConcerning the management of myelolipomas, surgery is indicated in the presence of tumor-associated symptoms with a large-sized mass, suspected malignancy, and tumor growth or hemorrhage . HoweverThe management of simultaneous adrenal and extra-adrenal myelolipoma is more challenging. One reason is the extreme rarity of these cases. So, the management must be considered on a case-by-case basis. Our report and the report by Zieker et al.  show thaIn our case, because the mass was preoperatively considered to be a single bi-lobulated myelolipoma, we scheduled a laparoscopic surgery due to the large size of the mass and the bi-lobulated appearance of the mass, considering the probability of separate masses. And surprisingly, two masses were seen during the operation. After adrenalectomy, the adrenal mass was observed to be smaller than the reported size in the preoperative CT scan. So, the retroperitoneal space was explored, and a retroperitoneal mass was found. Based on the lack of suspected malignancy and the appearance of the retroperitoneal mass, which was round, well-differentiated, and utterly similar to the excised adrenal mass, no intraoperative frozen section was sent. In the case by Zieker et al. , the manAs the presence of simultaneous adrenal and extra-adrenal myelolipoma is extremely rare, its management is more challenging than a single mass. It should be considered as one of the differential diagnoses. However, because this situation is extremely rare, the probability of malignancy, especially retroperitoneal liposarcoma, should be highly considered, and we suggest an obsessive approach when approaching this condition. It is important to manage these cases on a case-by-case basis and tailor the management concerning intraoperative biopsy, the intraoperative appearance of tumors, and the location of extra-adrenal masses. Of note, as this study is only a case report, maybe, the proposed algorithm cannot be generalized to all patients presenting with simultaneous adrenal and extra-adrenal myelolipoma in the future. Therefore, future reports may help the literature on the approach to simultaneous adrenal and extra-adrenal tumors, including myelolipomas.


A salivary mucocele is the most common disorder affecting the salivary glands. Saliva leakage from the salivary gland parenchyma and/or from associated duct damage causes a chronic inflammatory process which can occasionally result in osseous metaplasia. Dogs having an ossified sialocele present with a hard, non-fluctuating mass containing a viscous, sticky liquid at centesis. Either radiography or computed tomography can confirm the thick bone-like wall pseudocyst. Surgical excision of both the pseudocyst and the affected salivary gland represents the therapeutic gold standard in these cases. The aim of this study was to report the clinical and diagnostic findings, surgical management and histopathologic report of three cases of cervical sialocele complicated by pseudocapsule osseous metaplasia.fibrodysplasia ossificans progressiva. The aim of the present case series was to describe three dogs affected by a calcified salivary mucocele. The medical records of dogs affected by a cervical sialocele were retrospectively evaluated, and three cases met the inclusion criteria. All the dogs in this study were referred to the Veterinary Teaching Hospital (VTH) of the Department of Veterinary Sciences of the University of Turin  for a large solid mass in the intermandibular region. The diagnosis of a mucocele was confirmed clinically by centesis and by radiography or CT. Complete excision of both the pseudocyst and the ipsilateral mandibular/monostomatic sublingual salivary gland was performed in all cases. The histological report showed large areas of bone metaplasia within the pseudocapsule and chronic sialadenitis. Based on this limited case series, complete excision of the pseudocyst and a concurrent sialoadenectomy provided an effective treatment for this rare salivary mucocele disorder.Saliva is an irritant of the subcutaneous tissue, thus causing the development of a non-epithelial reactive pseudocapsule. Metaplastic ossification of the pseudocapsule is a condition rarely described in the veterinary literature. The main causes of calcification are trauma, tumours, various chronic inflammatory conditions and  Salivary gland (SG) disorders have rarely been described in dogs and cats, with only a 0.3% overall prevalence of veterinary consultations . SalivarThe clinical presentation depends on the location: A cervical sialocele usually appears as a slowly enlarging nonpainful swelling in the intermandibular area. It can sometimes be difficult to differentiate the side of the affected gland involved in sialocele; if located on the midline, it may only slightly shift toward the originating side in a patient placed in dorsal recumbency. Centesis usually reveals a transparent viscous mucoid fluid, sometimes slightly bloody and/or cloudy. Periodic Acid–Schiff (PAS) staining can confirm this fluid as being saliva by revealing mucin ,12.A sialocele can be investigated using radiography/sialography, ultrasound, magnetic resonance imaging (MRI) and computed tomography (CT) ,14,15,16Metaplastic ossification within the sialocele, for the most part at the level of its capsule, has previously been reported ,20,21,22The pseudocapsule of a salivary mucocele is microscopically visible as granulation tissue enclosing mucin; it is made up of edematous, vascularised connective tissue containing macrophages, lymphocytes and other chronic inflammatory cells.In human medicine, an oral sialocele is a common SG disorder which appears more often on the lower lip mucosa or mandibular vestibule and is frequently caused by accidental lower lip biting. Bone metaplasia has also been reported in individuals with a caecal appendix ,24 and aIn the past, the treatment of sialocele with drainage alone was characterized by a high rate of recurrence . SurgicaNo cases of human oral ossified sialocele have been reported to date. In veterinary medicine, only four case reports of sialocele-associated osseous metaplasia have been described ,20,21,22The medical records of dogs referred to the Veterinary Teaching Hospital (VTH) of the Department of Veterinary Sciences of the University of Turin  for sialocele from January 2000 to January 2023 were identified and reviewed.The inclusion criteria for this case series were as follows: (a) the presence of a mandibular/neck hard swelling; (b) a first or second level of diagnostic investigation to confirm the presence of a calcified lesion; (c) a sialoadenectomy, and complete or partial removal of the calcified lesion; (d) a complete histological report and (e) a minimum 30-day follow-up.Written consent was obtained from the owners to proceed with anaesthesia and all the diagnostic and therapeutic procedures.Perioperative standard-of-care management, including analgesia, was performed in all dogs. This study did not fall within the application areas of Italian Legislative Decree 26/2014, which governs the protection of animals used for scientific or educational purposes. Therefore, ethical approval was waived for this study. The animals were not treated as part of an experimental study; only the data were later selected and included in this study. No specific informed consent statement was obtained for inclusion in this retrospective study.®, Orion Pharma, Turku, Finland) and methadone (0.2 mg/kg) . General anaesthesia was induced using propofol   and was maintained with isoflurane in oxygen . The CT images were acquired in basal condition and after the intravenous administration of contrast medium Iomeron 400 mg/mL (1.5 mL/kg IV) .The head and neck were investigated using CT  or radiographically . For the CT scan evaluation, the dogs were premedicated with an intramuscular combination of dexmedetomidine (2 mcg/kg)   was performed 20 min before skin incision and was repeated every 60 min until extubation. The affected SG was removed together with the entire ossified lesion. The suction of saliva was performed using a Poole suction tube. Both SGs and the pseudocapsule underwent histopathology; however, a portion of the pseudocapsule also underwent microbiological analysis. A passive drain was inserted at the surgeon’s discretion.Both SGs and the pseudocapsule were fixed in 10% buffered formalin, decalcified for 48 h in Osteodec , embedded in paraffin, sectioned at 4 μm and routinely stained with haematoxylin and eosin. A histological diagnosis was reached by the pathologists of the Department of Veterinary Science, University of Turin.®, Eurovet Animal Health B.V., Bladel, The Netherlands; 0.2 mg/kg IV q4h) or buprenorphine ; and meloxicam . The dogs were discharged within 1–5 postoperative days with an Elizabethan collar and meloxicam , and were then re-evaluated weekly for a month after discharge. Complications were classified according to the guidelines of the “Veterinary Co-operative Oncology Group—Common Terminology Criteria for Adverse Events” (VCOG-CTCAE) [Postoperative management included intravenous fluid (Lactated Ringer’s 2 mL/kg/h); analgesia with methadone (SemfortanG-CTCAE) . The skiThree dogs presenting an ossified sialocele met the inclusion criteria and were included in this case series.A 3-year-old, 28 kg, intact female Boxer was referred to the VTH for a 6 cm painless firm swelling at the level of the right ventrolateral region of the neck. The swelling had been present for 3 months and had gradually grown. The dog showed no clinical signs of pain or discomfort except for drooling. Both the physical examination and laboratory profile  were within normal limits. There was no clinical evidence of regional lymphadenomegaly.Sterile needle centesis was performed and revealed the presence of a stringy, brown fluid. Cytology of this fluid was consistent with saliva, owing to the presence of mucin aggregates, with no evidence of infection. Radiographic examination of the head and neck showed a large soft tissue consolidation with a calcified wall between the right intermandibular and subauricular regions. A skull CT scan was performed to identify the origin of this consolidation and its anatomical relationship with the surrounding structures. The examination revealed an ovoid capsulate mass (3.6 × 4.7 × 4.3 cm) at the level of the right pharyngeal region . The capThe dog was placed in left lateral recumbency, and a rolled towel was placed under the neck to elevate the surgical site. A lateral surgical approach to the right mandibular SG was performed. The cystic mineralised lesion was operated on using a 10 cm skin incision, from the ventral aspect of the vertical ear canal to the caudal portion of the ramus of the mandible, just over the swelling. The platysma and parotidoauricularis muscles were incised and reflected ventrally. The lateral portion of the calcified lesion was gently isolated from the surrounding tissue and completely excised .Part of the capsule and its contents were sent for microbiological analysis. The right medial and lateral retropharyngeal lymph nodes were excised.Subsequently, a mandibular/sublingual SG and duct sialoadenectomy was performed; the duct was ligated and transected as rostrally as possible at the level of the lingual nerve, which was spared. Using a combination of blunt and sharp dissection, the glandular tissue and its duct were dissected from the capsule and excised by tunnelling under the digastricus muscle. The surgical site was flushed with warm saline solution and a Penrose drain was placed within the subcutaneous space exiting ventral to the incision. The incision was closed routinely in three layers. Part of the capsule, the excised SG and lymph nodes were sent for histological evaluation.The dog recovered uneventfully within a week. The bacteriological analysis of the pseudocapsule was negative for any bacterial growth.Histologically, the SGs showed multifocal moderate lymphoplasmacytic chronic sialoadenitis with the interstitial spaces expanded by thick bundles of fibrocytes (fibrosis). The cystic lesion was lined by granulation tissue and small foci of an osseous metaplasia . The lymThere was no sign of recurrence more than five years after surgery.A 1.5-year-old, 38 kg, intact male Italian Cane Corso was admitted for an 8 cm hard swelling just caudal to the left mandibular ramus, which had been present for 6 months and significantly increased in size during the previous 2 weeks. The consistency appeared solid at physical examination. The macroscopic aspect of the fluid obtained by centesis was compatible with saliva; however, cytology was inconclusive. The latero-lateral radiographic view of both the skull and the cervical region highlighted an irregularly rounded area of bone-like radiopacity at the level of the pharyngeal region superimposed to the hyoid apparatus . Both thBased on these findings, a suspicion of sialocele was formulated and a left mandibular/sublingual SG and duct excision was proposed. The patient was positioned in right lateral recumbency. A rolled towel was placed under the neck to elevate the surgical site. A vertical incision centred on the ossified lesion was performed caudally to the angular process of the mandible . The subA 12-year-old, 8 kg, spayed female Dachshund was referred for a 5 cm painless swelling in the intermandibular region. The lesion had grown during the previous two weeks and appeared firmly attached to the surrounding tissues. The lesion had a bony/cartilaginous consistency; however, its ventral border appeared incomplete as several small openings were palpated. Due to the compression on the pharynx and the trachea, the dog presented with dyspnea on exertion. A small right sublingual sialocele was also present. Haematologic laboratory tests showed increased gamma-glutamyl transferase, bilirubin, total protein and hypoalbuminemia. These anomalies, associated with potbelly and bilateral and symmetrical back alopecia, were suggestive of hyperadrenocorticism, and an internal medicine specialist consultation was required. Low dose dexamethasone suppression test confirmed Cushing’s syndrome. Cytological analysis of the swelling was compatible with saliva; therefore, a calcified sialocele was clinically suspected.A CT scan revealed a complex cystic formation extending from the right mandibular region, between the tongue and the mandible, up to the ipsilateral laryngeal region. The swelling had dimensions of 4.5 cm (width) × 5.8 cm (height) × 6.1 cm (length) with multiple concamerations and partially mineralised walls (approximately 2 mm thick); the content was fluid attenuating with no contrast enhancement (average attenuation of 20 HU). The medial aspect of the lesion was in contact with the hyoid apparatus and the larynx, which were dislocated and compressed to the left, while the caudal margin was in contact with the right mandibular lymph node and the facial vein. The swelling extended dorsally in the pharyngeal region, reaching the right tympanic bulla and the external carotid artery. The right mandibular SG appeared adherent to the lateral wall of the swelling, resulting in its appearing dislocated and compressed laterally. The right retropharyngeal lymph node was enlarged (2.2 × 1.9 cm) with dishomogeneous contrast enhancement and was in contact with the caudal aspect of the swelling .The adrenal glands were enlarged bilaterally. The right measured 2.2 cm × 1.1 cm and the left 2.3 cm × 1 cm with dishomogenous contrast enhancement. No macroscopically visible pituitary gland abnormalities were found.®, Dechra Veterinary Production S.r.l., Northwich, UK) therapy at 1 mg/kg q24h was started. On the day of surgery, the dog was positioned in dorsal recumbency, with a towel placed under the neck to slightly elevate it. A skin incision in the intermandibular region centring on the ossified lesion was performed. The platysma muscle was incised, and the ventral aspect of the lesion was separated from the surrounding tissues  in relation to the position of the SGs should be evaluated. The suspected SG is generally contiguous to the fluid collection, with the affected gland which may appear atrophied, normal or enlarged at CT. The attenuation of SGs at CT may be normal or altered, sometimes with fatty infiltration or fluid pockets. These findings, associated with the presence of local fat stranding, may help in identifying the SG involved in the sialocele. Moreover, the need for advanced imaging in a preoperative setting is confirmed by the anatomical variations of the mandibular SG reported in the recent literature . AlthougIn a sialoadenectomy and duct excision regarding sublingual and mandibular SGs, both ventral and lateral approaches can be performed . The venIn the literature, a clear recommendation regarding the complete excision of the pseudocapsule is lacking ,52. In tIn all the dogs, the bacteriological analysis carried out on a sample of the pseudocapsule and its contents was negative, thus confirming the non-infectious origin of the lesion. Histological examination was performed in all cases, and it excluded a neoplastic process affecting the SG but, on the contrary, confirmed its inflammatory origin. Finally, the decision to perform an excisional biopsy of the tomographically or clinically enlarged lymph nodes can be a critical point; it was dictated by the fact that the unusual sialocele clinical presentation without the definitive histological diagnosis could not totally exclude a tumour.The lymph node excision did not require additional skin incisions. The excised lymph nodes were adjacent to the glandular structures removed; therefore, their dissection did not lead to lengthening the surgical time or to an increase in post-surgical complications . HyperplThe main limitations of this study were its retrospective nature and the small number of cases included, the latter reflecting the rarity of this condition. Another limitation was the different method of diagnostic imaging utilised as, for patient #2, a CT scan was not available.The evidence of a huge and consolidated mass at the level of the neck should not immediately be considered highly malignant, and a complex sialocele should be considered in the differential diagnosis. Cytology, centesis and imaging are necessary to achieve the diagnosis and to plan the surgical approach. Ossified sialocele excision is associated with a favourable prognosis if the affected SGs are removed together with a part of or the entire pseudocapsule. Definitive diagnosis is provided by the histological examination.


Placenta percreta is the most severe and least common form of placental insertion abnormalities. The increasing frequency of C-Section deliveries has led to more of these abnormalities. Ultrasound and magnetic resonance imaging (MRI) have a key role in diagnosing these abnormal adherences since it shows best transmural extension of the placental tissue. We report a case of a woman with a previous cesarean delivery who had been diagnosed with a placenta preavia on ultrasound and a suspicion of transmural extension with her MRI later showing a placenta percreta. Due to the increasing number of cesarean deliveries, abnormal placental insertion has also increased in frequency becoming one common cause of postpartum hemorrhage and peripartum morbidity. It includes three grades based on the severity of placental invasion: increta, accreta and percreta. The latter is a term given to the most severe but least common form of the spectrum of abnormal placental villous adherence, where there is a transmural extension of placental tissue across the myometrium with a serosal breach. This extension can only be seen on ultrasound and magnetic resonance imaging (MRI) that highlights the role of the radiologist in the diagnosis.We report the case of a 30-year-old woman who had a C-Section 2 years earlier for fetal macrosomia. She presented in our department for a normal routine fetal ultrasound that identified some intra-placental lacunae as well as the protrusion of placental tissue beyond the uterine myometrium, with a discontinuity in the urinary bladder wall and the extension of Doppler vascularity between serosa and the bladder . AbnormaHer MRI showed some thick linear and round bands arising from the maternal side of the placenta seen in low signal in single-shot fast spin echo (SSFSE) T2-Weighted images (HASTE) and in high signal on Balanced Steady State Free Precession (bSSFP) (True Fisp), corresponding to dark intra-placental bands . The mosPlacenta percreta is the most severe, yet least common, form of abnormal placental insertion  . It is dHistopathological examination remains the main exam to confirm the diagnosis, but unfortunately, this can only be confirmed in the event of hysterectomy, while nowadays conservative treatment is favored .Normal placenta echo structure changes during pregnancy. In the second trimester, it is homogenously hyperechoic while in the third trimester, it turns to a more heterogeneous calcified aspect associated to physiological placental lakes  . On the In the case of placenta percreta, ultrasound may show the loss of retroplacental clear space, a reduced myometrial thickness as well as placental lacunae. The latter finding corresponds to parallel linear vascular channels extending from the placental bed into the myometrium. These structures tend to show turbulent flow in Doppler and ‘Swiss cheese’ aspect (if there are many of them) . These let al. published in 1992 the first case of MRI diagnosis of placenta percreta on a former placenta previa [Ultrasound in diagnosing placental percreta by ultrasound has its limitations, which has led to introducing the MRI as complementary imaging tool. Thorp a previa .T2-WI are taken in the three planes: strict sagittal, axial and coronal. Placental invasion is best seen using T2-weighted sequences without fat suppression. bSSFP sequences (named True Fisp on Siemens and Fast Imaging Employing Steady-state Acquisition (FIESTA) on General Electric (GE)) and fast sequences (such as SSFSE on GE and Haste on Siemens) have a huge advantage of being very little artefacted by maternal and fetal movements .et al. suggested performing it before 35 weeks of pregnancy while Lim et al. proposed before 30 weeks [nd trimester is a homogeneous isosignal aspect respecting the myometrium on T2-WI. As for the 3rd trimester, the placenta begins to have a more T2 hyposignal lobular and heterogeneous aspect. The myometrium usually has high signal on T2-WI , we might have dark intra-placental bands on T2-WI with a heterogenous appearance of the placenta and abnormal disorganized placental vascularity seen as traversing vessels. There’s also focal interruptions of the myometrial wall and the most particular sign in placenta percreta is the tenting of urinary bladder with the involvement of uterine serosa or extra-uterine involvement of adjacent organs like bladder, rectum or abdominal wall  Fig. 5)Fig. 5).This uncommon pathology requires a precise diagnosis in order to guide the right obstetrical care. Surgical intervention is the pillar of treatment in most cases, but bleeding during the intervention is a serious concern particularly in situations where the bladder or (other adjacent structures) are involved. Imaging remains key in diagnosing placenta percreta with ultrasound being the first-line examination and MRI coming in second when the diagnosis is uncertain or when the myometrial wall extension assessment is incomplete on ultrasound.El Harras Yahya was responsible for manuscript concept, design as well as editing and literature search. Safaa Choayb helped in manuscript editing and literature search, manuscript editing and manuscript review. Najlae Lrhorfi contributed to conception and design. Nazik Allali contributed to acquisition, analysis and interpretation. Latifa Chat critically revised the manuscript and gave final approval. Siham El Haddad contributed to acquisition, analysis and interpretation; critically revised the manuscript and gave final approval.The authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper.This research received no specific grant from any funding agency in the public, commercial or not-for-profit sectors.


Metanephric adenoma is a rare benign renal tumor of the kidney, uncommonly observed in children. It is often misdiagnosed preoperatively as a malignant neoplasm, leading to an unnecessary nephrectomy. The challenge is to make the right diagnosis preoperatively and therefore manage it with conservative surgery. We report a case of a child with metanephric adenoma who underwent nephron-sparing surgery.A renal tumor was discovered fortuitously in an 18-month-old Caucasian girl with several congenital malformations. Investigations showed a 28 × 27 × 27 mm left renal mass centrally located, well defined, nonvascularized, with no calcifications and which compressed the adjacent renal tissue. Furthermore, there were no signs of metastasis. The decision of a multidisciplinary meeting was to perform a computed tomography (CT)-scan-guided biopsy. Histologic examination concluded it was a metanephric adenoma. We performed a left open partial nephrectomy via a flank retroperitoneal incision. The final histopathological examination confirmed the diagnosis. The postoperative course was uneventful.Preoperative diagnosis of metanephric adenoma is challenging. Because of the high probability of unnecessary radical nephrectomy, preoperative biopsy can be safe and determining to guide a more conservative approach so nephron-sparing surgery can be performed. Metanephric adenomas (MAs) are rare benign neoplasms of the kidney. They derive from the renal residual tissue during embryonic development. These tumors occur predominantly in middle-aged women with few cases reported in children , 2.Because of the lack of specific clinical and radiographic characteristics, they are frequently misdiagnosed preoperatively as malignant tumors of the kidney. The diagnosis is mostly established based on histological examination after an unnecessary radical nephrectomy. However, accurate diagnosis made preoperatively is of great importance as MA should be treated conservatively with nephron-sparing surgery. Herein, we describe the case of an infant who underwent conservative treatment for a metanephric adenoma diagnosed by preoperative fine-needle biopsy.An 18-month-old Caucasian girl was referred to our department after the fortuitous discovery of a renal mass. She presented with history of microcrania, cataract, and strabismus with no specific malformative syndrome. There was no family history of consanguinity, malformative or oncologic disease. She was operated on for her congenital cataract with uneventful course. She was treated by valproic acid for one episode of idiopathic epileptic crisis. On initial physical examination, hemodynamic constants and temperature were normal, we found a microcranium with cranial perimeter of 41 cm under 3 Standard Deviation (SD)  with nothing to notice on the rest of the neurological examination. There was also no abdominal tenderness nor a palpable mass. On biological assessment: hematology, electrolytes, renal, and hepatic functions were normal aside from a polycythemia. The activated partial thromboplastin time (APTT) or activated clotting time (ACT) was extended with a value of 63 s, prothrombin time was at 100%, and Rosner index was at 16.43%, which could be due to a lupus anticoagulant, although specific investigations ruled out this probability. Urine cytology and culture results were within normal limits, without hematuria. As part of an assessment for other malformations, an abdominal ultrasound was performed, which showed a mass arising within the left kidney.Abdominal ultrasonography revealed a small left round solid central renal mass, hyperechoic, well defined, weakly vascularized without calcifications. It measured 26 × 28 mm.CT scan of the abdomen showed a small round subcapsular tumor, arising from the medial part of the left kidney (28 × 27 × 27 mm). It was well defined and compressed the adjacent renal tissue. The tumor was not clearly enhanced in the early phase on CT. There were some areas of necrosis, but there was no evidence of metastatic disease Fig. . Wilms’ A left flank retroperitoneal incision exposed the left kidney. The inspection showed a well-circumscribed nonencapsulated polylobed tumor. Its appearance was similar to that of normal kidney tissue. The tumor measured 30 × 20 × 30 mm with a noticeable cleavage plane. An easy wedge resection of the tumor was performed using the electric scalpel, ensuring perfect hemostasis and respecting the caliceal cavities.Macroscopically, the tumor was well circumscribed and yellow–white and had an intact tegument with homogeneous and gray cutting surface Fig. . MicroscOncologists and surgeons recommended no further treatment. The patient had an uneventful postoperative course and was discharged on the fourth day. The patient was followed postoperatively by physical examination, abdominal ultrasound, and biological renal function every 3 months during the first year, and then every 6 months. The clinical, biological, and radiological follow-up was uneventful 4 years later.Management of renal masses in children can be challenging, especially when lacking specific radiologic features. MA is a less common benign condition in children; its similarity to other malignant renal tumors makes establishing a preoperative diagnosis very important to guide the adequate surgery technique. To our knowledge, our patient is the youngest case of a child with MA who underwent nephron-sparing surgery .et al. [et al. [Clinically, MA may present with hematuria, flank pain, hypertension, or abdominal mass –5. Howev [et al.  reported [et al. . Thus, i [et al. .The final diagnosis is confirmed by postoperative histopathological examination, which accurately describes all the characteristics of the tumor . MicroscPartial or radical nephrectomy is the mainstay of treatment for MA. Radical nephrectomy still remains the treatment of choice when the preoperative biopsy is not conclusive.The possibility of nephron-sparing surgery in MA in the pediatric population has previously been addressed in literature . Partialet al. [The follow-up is not well defined, but should be done with clinical and radiological examination according to Liniger et al. .MA is rare in children, and even rarer in infants. Its preoperative diagnosis is challenging as it lacks specific imaging features. Giving the high probability of subjecting infants to an unnecessary radical nephrectomy, preoperative biopsy can be a safe procedure that guides the management plan toward the more conservative nephron-sparing surgery.


Dyke-Davidoff-Masson syndrome (DDMS) is a rare congenital or acquired neurological disorder that most commonly affects the pediatric population but is also rarely reported in adults. DDMS results from brain injury in the intrauterine or early years of life. It is characterized by prominent cortical sulci, hyperpneumatization of the frontal sinus, unilateral cerebral hemiatrophy with ventricular dilation, and associated bony thickness of the cranial vault. Seizures and asymmetric hemiparesis are the most consistent findings in DDMS with facial asymmetry and mental retardation widely reported. Herein, we report a case of a 32-year-old female patient with DDMS presenting with a history of seizure and right-sided hemiparesis. Neuroimaging findings showed asymmetric cerebral encephalomalacia and gliosis with ex vacuo ventricular dilatation and calvarial diploic space widening. Our case report is unique in the sense that our patient presented with DDMS in adulthood with no signs of mental retardation or history of seizures during childhood and well-controlled seizures on monotherapy. Given the adult presentation of DDMS is unusual and rarely reported in the medical literature, our case report will help physicians to keep DDMS high on differential diagnoses in such cases. Awareness of the clinical features of DDMS on imaging can facilitate a timely and accurate diagnosis, thereby enabling appropriate and prompt management. Dyke-Davidoff-Masson syndrome (DDMS) is a rare congenital, neonatal, or early infantile condition which classically involves cerebral atrophy or hypoplasia. It usually occurs due to an insult to the developing brain in intrauterine or early childhood . There aDDMS frequently manifests in males and involves one cerebral hemisphere, predominantly the left cerebral hemisphere. The age of presentation depends on the time of the neurological insult, with most cases being diagnosed in either childhood or adolescence due to predominant clinical manifestations. Up until 2018, only 21 cases have been reported where the disease was diagnosed in adulthood . An exteDDMS has a wide spectrum of clinical presentations. Classically, the disease presents with unilateral upper motor neuron lesion-like findings  such as hemiparesis or hemiplegia, a variable degree of facial asymmetry, seizures, mental retardation, and behavioral changes . SensoryDiagnosis of DDMS is based on the combination of clinical and imaging findings. The diagnostic workup includes electroencephalography (EEG) (in patients who present with seizures) and radiographic studies using brain CT scans and MRI, which are the gold standard. Early manifestations of the disease are often missed on brain CT, and therefore, an MRI brain needs to be done in all patients with suspected DDMS . Brain MNo standardized management of DDMS exists, and it is mostly symptomatic. The disease management for adult-onset DDMS is different from childhood DDMS . InterveWe present the case of a 32-year-old female who presented to our medicine outpatient department (OPD) with a history of an episode of generalized tonic-clonic seizure (GTCS) in the past 24 hours. She initially had an episode of an unprovoked seizure one year back, following which she was on Levetiracetam but discontinued the medication a month ago. The patient’s past medical history was significant for a stroke resulting in right-sided hemiparesis at the age of 2.5 years, with residual right-sided weakness. Upon admission to the medicine ward, the patient was provisionally diagnosed with a seizure disorder.The patient’s vital signs were within normal limits. She was conscious and oriented. Her higher mental function was normal, and her sensory function appeared to be intact. However, she displayed reduced bulk and power on the right side, as well as hypertonia and a plantar extensor response on the same side. In addition, she exhibited a circumduction gait. Brain CT scan results demonstrated left temporoparietal and occipital gliotic changes, likely related to the patient’s prior stroke, along with ex vacuo dilatation of the ipsilateral ventricle Figure . Further evaluation with magnetic resonance imaging (MRI) of the brain revealed encephalomalacia and gliosis in the left temporoparietal and occipital lobes with ex vacuo dilatation of the lateral ventricle and calvarial diploic space widening in the left parietal bone and the occipital bone, as shown in Figure The patient’s routine blood workup revealed an unremarkable lipid profile along with normal liver and kidney function tests. A sickling test was performed on the blood sample using high-performance liquid chromatography (HPLC), which yielded a negative result. The significant findings of the blood investigation are mentioned in Table Soon after discharge the patient returned to a private clinic for a follow-up appointment and exhibited progress. The only reported symptom was a mild headache and the patient was stable overall. It was noted that she had been consistent in taking her prescribed medications as directed.DDMS was first reported in the literature in 1933 by Dyke, Davidoff, and Masson when they studied a series of nine patients with unique disease features. A literature search showed the predominance of DDMS in childhood ,9,13. OnIn contrast to most cases of DDMS that present with a history of multiple seizure episodes ,15, our Our patient was diagnosed with DDMS using a brain CT scan, which showed left temporoparietal and occipital gliotic changes, and a brain MRI, which is of great importance in DDMS diagnosis, which showed gliosis in the left temporoparietal and occipital lobes, along with ex vacuo dilation of the left lateral ventricle and widening of the diploic space in the parietal and occipital bones. This was consistent with the radiologic findings found in many other patients with DDMS . A studyThe dilemma is that there are no definitive treatment guidelines for seizure management in DDMS patients. Various antiepileptic drugs are used either alone or in combination. Patients have responded differently to different antiepileptics. Bhol et al. described two cases where both DDMS patients with focal seizures responded differently to the same anticonvulsant therapy . Al-SmaiBecause of its clinical uniqueness and wide spectrum of presentation, the prognosis of DDMS is unclear and depends on how early the disease is diagnosed. Early recognition of the disease with proper intervention has a better prognosis . The disDDMS is a neurologic disorder characterized by a constellation of symptoms of varying severity. Adult presentation of DDMS is a rare finding with no relevant pediatric history. As this is a very rare disorder, it can easily go undiagnosed by the inexperienced eye. Management is variable and depends on the age of the presentation and the severity of the symptoms. This case report of an adult patient with DDMS with a history of seizure and hemiparesis and no other significant findings of DDMS emphasizes the importance for physicians to consider DDMS as a potential diagnosis in such cases, in addition to managing the seizures with monotherapy. Powerful imaging studies like brain CT and MRI are thoroughly advised when attending to patients with clinical features like DDMS to confirm the diagnosis. Larger longitudinal studies are advised to better understand the management options for DDMS and the subsequent prognosis.