Heterotopic ossification (HO) histologically refers to extraskeletal bone formation in non-ossifying tissues, most commonly noted in the extremities, buttocks, abdominal wall, and hip joints. HO developing in the mesentery  is very rare, with fewer than 100 cases reported in the literature. It usually occurs in adult male patients with a history of repeated abdominal trauma. So far, only two cases of HMO have been reported with the development of hematopoietic bone marrow. Here, we report the third case of HMO with true trilineage hematopoiesis in a 66-year-old female with suspicious mesenteric-retained foreign material from prior surgical procedures, including hysterectomy for endometrial adenocarcinoma and multiple repairs for incisional hernia. Heterotopic ossification (HO) refers histologically to extraskeletal bone formation in non-ossifying tissues with a yet to be determined incidence, exact etiology, and pathogenesis -4. It wa6/µL; hemoglobin, 7.6 g/dL; hematocrit, 23.5%; mean corpuscular volume, 81.6 fL; mean corpuscular hemoglobin, 26.3 pg; mean corpuscular hemoglobin concentration, 32.2 g/dL; red cell distribution width, 15.4%; platelet count, 145 k/µL, mean platelet volume, 8.2 fL). Computed tomography of the abdomen reported a linear opacity adjacent to the gastric body and antrum, as well as a high-density mass in the mesentery suspicious for possibly retained foreign body. After five abdominal surgeries for a complicated postoperative course, she fully healed and underwent an ileostomy take-down one month later. During the surgery of ileostomy take-down, the mesentery appeared markedly congested and slightly adhered to the anterior abdominal wall. An intra-abdominal mass was palpated in the mesentery. Pathological gross examination showed that the excised mass was well-circumscribed and firm, located in fibrous and adipose tissue, and measured 1.5 cm in the greatest dimension with a whitish trabeculated cut surface. Microscopic examination showed mature cortical lamellar bone with osteocytes, cement line and rimming osteoblasts, and true trilineage hematopoiesis with erythropoiesis, granulocytopoiesis, and megakaryocytopoiesis  for both HMO and ectopic marrow development remain to be elucidated.


Osteoid osteomas (OO) are benign bone tumors that are prevalent in young adults. The typical clinical picture of the disease is pain that worsens at night, which may be alleviated by Non-Steroidal Anti Inflammatory Drugs (NSAIDs). The most common imaging finding of OO is a lytic lesion, known as a nidus, with variable intralesional mineralization, accompanied by bone sclerosis, cortical thickening and surrounding bone marrow edema, as well as marked post-contrast enhancement. The most commonly affected sites are the long bones of the lower limbs, but the ribs are rarely reported sites. The present study describes a case of osteoid osteomas located in the rib which was removed by rib resection using CT-scan guidance. •Osteoid osteoma (OO) is a benign bone tumor and the most commonly affected sites are the long bones of the lower limbs, but the ribs are rarely involved.•Careful history taking, meticulous physical examination, and investigations are necessary.•Prompt treatment should be addressed in a proper way to stabilize the patient and avoid mortality. OO is diagnosed by both clinical and imaging findings. A biopsy is recommended, especially for lesions with an atypical presentation, even though it can be non-diagnostic in nearly one-third of cases Herein, we report a rare case of a 13-year-old male diagnosed with osteoid osteoma (OO) of the ninth rib after presenting as continuous chest pain that is worst at night with a known history of trauma at the same site of the tumor. This work has been reported in line with the SCARE criteria, which are used by authors, journal editors, and reviewers to increase the robustness and transparency in reporting surgical cases 2visual analog scale (VAS). The pain was refractory to Non-Steroidal Anti Inflammatory Drugs (NSAIDs) and acetaminophen prescribed by his family physician. There were no associated symptoms including cough, shortness of breath, or increased sputum.This is a 13-year-old male known case of asthma, referred to the thoracic surgery department complaining of chest pain for two months duration. The pain was primarily right-sided, anterolateral aspect of the lower chest. It was continuous all over the day, and become more severe at night, measured at 07/10 according to the One year ago, there was chest trauma over the same location. At that time, the patient was seen in the emergency department (ED) with 5/10 right-sided chest pain. After that, a Chest X-ray was performed and showed no abnormalities . The patUpon examination, he was in a good general condition and normal vital signs. A fixed tenderness point was identified on the anterolateral aspect of right lower chest over the ninth rib at the level of the anterior axillary line. No associated erythema or increase in temperature. Chest bulge or deformity was also absent.CT scan  to mark the lesion, the lesion was identified and marked using a skin marker, the lesion itself was marked, and 4 cm margins were identified medially and laterally to the lesion.Under general anesthesia, Double lumen intubation was performed with an isolate of the left lung. Arterial line and Folly's catheter were inserted, and a thoracic epidural catheter was applied for postoperative pain management. The patient was placed in the left lateral decubitus position, an anterolateral thoracotomy was performed. After identifying the nidus with a marker using CT scan guidance, a right-sided chest 10-cm incision parallel to the 9th rib was made. Complete resection of the lesion with a wide, clear safe margin was followed which includes about 4 cm medial and lateral to the lesion. In addition, the overlying serratus anterior muscle was also resected, because it was invaded by the lesion too, with attention paid to avoid an injury to the neurovascular bundle. A 20-French straight chest tube was placed and secured. The surgical incision was closed in layers. Complications were not observed during the operation. The bone specimen was sent for histological study.2.2The tumor is well circumscribed and formed of haphazardly arranged trabeculae of woven bone, which rimmed by osteoblast in a loose vascularized stroma  makes up about 10 % to 15 % of the total. It primarily affects young people, primarily in a male between the ages of 5 and 25 Only 5–7 % of all primary bone neoplasms are rib-specific primary tumors The diagnosis of OO is made using a mix of common clinical images and imaging results. The primary nocturnal symptom of this illness is pain, which can be treated with salicylates and non-steroidal anti-inflammatory medications (NSAIDs). The nidus can be hidden on radiographs; hence the CT is seen to be the best modality for OO. The central calcification is typically regular and centered, but it can also be punctate, amorphous, or ring-like. A “vascular groove” or “CT vessel” sign that corresponds to larger vessels that emerge from the periosteum to irrigate the hypervascular nidus can be seen on CT scans For the treatment of OO, both conservative and surgical methods have been reported. A long-term NSAID regimen is used as conservative treatment, and in some circumstances, it is said to help people feel less pain. Incomplete nidus excision could result in local recurrence, according to the general principle for surgical therapy of OO. When the afflicted bone is not subcutaneous or when the visual localization of the nidus is technically challenging, a full resection of the nidus by open surgery may result in additional harm to the bone and soft tissues surrounding the lesion.Although it is simple to access the ribs, the reported lengths of en bloc nidus resection ranged from 5 to 9.5 cm, which is much wider than the nidus's size 4This case study demonstrates that osteoid osteoma of the rib should indeed be recognized in cases of any painful rib without such a recent history of trauma and that total surgical excision, when possible, is a safe and successful treatment for this extremely uncommon bone tumor. In order to reduce the loss of respiratory function, CT-guided operations should be taken into consideration as the preferred course of treatment for OO of the rib.Informed consent was signed from the patient for publication.Study concept or design: Yousef Abu AsbehWriting the manuscript: Salem M. Tos, Afnan W.M. Jobran, Anas Alasafrah and Motaz NatshehReview & editing the manuscript: Salem M. Tos, Afnan W.M. JobranWritten informed consent was obtained from the patient and his parents for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.Dr. Yousef Abu Asbeh.Not applicable.The authors declare no conflicts of interest.


Unlike subcutaneous lipomas, thoracic cavity lipomas are extremely rare and can develop to be quite large without causing any symptoms. However, managing massive lipoma that involves both chest cavities is usually challenging, especially when considering the approach for excision.We report our experience of surgical management of a case of a 46-year-old male with huge intrathoracic lipoma that extends bilaterally and is known to be the largest of such kind. The tumor was resected successfully using median sternotomy. Histological analysis confirmed features of lipoma.To remove a bilateral intrathoracic lipoma, various surgical approaches have been documented. In our experience, a median sternotomy allows better exposure, which aids in complete surgical extirpation resulting in the prevention of recurrence. Lipomas are the most prevalent benign mesenchymal tumors, while intrathoracic lipoma is a rare entity. On the other hand, giant lipoma involving both hemithorax is far more unusual, with only a few occurrences documented in the literature –3. LipomSurgical removal is still the primary line treatment for intrathoracic tumors with a good outcome , 2. HoweHere, we present our experience of surgical management of a case of a 46-year-old male with huge intrathoracic lipoma that extends bilaterally, displacing the lung upwards. To our knowledge, this is the largest reported intrathoracic lipoma with the bilateral extension, which was removed successfully through standard median sternotomy.A 46-year-old hypertensive man presented to our outpatient clinic with a dry cough that had been bothering him for 3 weeks, as well as periodic shortness of breath with chest heaviness when exerted. He has no prior history of chest pain, hemoptysis, or weight loss, nor does he have any relevant medical history. His blood pressure is controlled with medication. He was a cook, non-alcoholic, non-smoker, and having a body weight of 65 kg for his 166 cm height.Physical examination of the patient revealed absent breath sounds along with dullness on percussion over both the middle and lower chest. Routine blood workup was within the standard limit except for mild anemia and raised ESR.1) of 1.2 L (42% predicted), forced vital capacity (FVC) 1.7 L (49% predicted) and FEV1/FVC ratio of 70%. Preoperative echocardiography showed no evidence of cardiac compression with an ejection fraction of 61%.A routine chest X-ray revealed a massive homogenous mass that occupied the lower two-thirds of both chests Fig. a. CECT rThe chest cavity was explored through a standard median sternotomy to provide complete access to both the pleural cavities and the mediastinum in order to remove the massive intrathoracic lipoma. The gross appearance was a thinly encapsulated, lobulated, dull mass with a pale yellow color that had a smooth surface and was soft to the touch Fig. . Mass waThe resected tumor weighed around 5400 g. It contains four lobulated parts, the largest of which is approximately 24*10*8 cm in size. The cut section comprises yellow lobulated fatty tissue  that creates obtuse angles with the chest wall and displaces neighboring pulmonary parenchyma and arteries on a chest CT permits a conclusive diagnosis 50 HU tha, 5, 11. When possible, total en-bloc excision of lipoma is the definitive treatment choice for preventing future recurrences. Complete surgical excision using the lateral thoracotomy or standard median sternotomy is the most applied surgical approach for intrathoracic lipoma . ClamsheIntraoperative problems are not infrequent because of severe adhesions with important structures caused by the mass's chronic existence in the thoracic cavity, as in our instance, where it was firmly connected to the innominate vein and superior venacva. Local recurrence of intrathoracic or mediastinal lipomas after resection is infrequent. They may, however, recur locally, and the risk of recurrence following an excision has been documented in the literature to be less than 5% . DespiteIntrathoracic lipomas are rare benign tumor in contrast of subcutaneous lipoma which is very common. They typically grow very slowly over years without any symptoms and signs. They often found in routine investigations incidentally when they become large. Whereas CT and MRI are usually diagnostic. Although pleural lipoma never evolves towards liposarcoma, surgical resection is still necessary and must be resected completely to prevent further recurrence.


The thrombosis of the superior mesenteric vein (SMV) can result in ischemia of the intestine. A 71-year-old male presented with pain in the abdomen and a black tarry stool. The findings from computed tomography (CT) with the contrast of the abdomen suggest the thrombosis of the SMV. Heparin was administered, followed by thrombectomy and stenting of the SMV. The patient reported no complications and was shifted from heparin to apixaban and then discharged after a five-day hospital stay. Acute mesenteric ischemia (AMI) is a sudden decrease in blood flow to a section of the intestine that causes ischemia, cellular damage, and intestinal necrosis . It requThe patient is a 71-year-old man who presented to the emergency department after experiencing increasing acute and severe atraumatic pain in the abdomen. He reported experiencing diffuse, generalized abdominal pain and black tarry stools for several months. He has a past medical history of two previous strokes. He reported seven months ago that he had an upper and lower endoscopy performed with findings of Barrett's esophagus, hiatal hernia, and hemorrhoids. He is currently on aspirin 81 mg every day (QD). He has a family history of strokes, which contributed to his father's death. He stated that he was a previous pack, a daily smoker for 50 years, and currently uses recreational marijuana. He denied extensive alcohol intake.On examination, no bleeding was present. Laboratory investigations revealed a white blood cell count of 13.5 K/µL, hemoglobin concentration of 13.6 g/dL, RDW of 14.6, PT of 10.8 seconds, INR of 1.0, and PTT of 24.6 seconds. Urinalysis was unremarkable, and COVID-19 RT-PCR was negative. A CT of the abdomen and pelvis with contrast was obtained and revealed SMVT with extensive perivascular infiltration with partial occlusion of the main portal vein (Figure Following the CT, interventional radiology was consulted. A portal venogram with thrombectomy, venoplasty, and stent placement of the SMV was performed without complications Figure . GastroeAMI is an abrupt cessation of blood flow to a portion of the small intestine that results in ischemia, cellular damage, and intestinal necrosis . AMI accThe SMV drains the intestine from the duodenum to the right two-thirds of the transverse colon and ends by joining the splenic vein to form the portal vein . SMVT deAlthough the incidence of SMVT is low, the mortality rate is high, i.e., 19-23% .Patients with acute and sub-acute MVT may present with abdominal symptoms like severe mid-abdominal pain, nausea, gastrointestinal bleeding, constipation, and fever due to phlebitis . AbdominPatients presenting with acute MVT are initially managed with analgesics, blood transfusion, and antibiotics . Then anOur case reports a 71-year-old male with a history of smoking and a family history of stroke. He presented to the emergency department with acute abdominal signs and a black tarry stool. The diagnosis of acute mesenteric ischemia due to SMVT was made based on the findings of the CT abdomen. Heparin therapy was started, and thrombectomy, venoplasty, and stenting of SMV were performed. The patient was shifted from heparin to apixaban and was discharged after a five-day hospital stay, during which he reported no complications. The patient was found to be stable on apixaban therapy two months after being discharged from the hospital.AMI accounts for a minimal number of acute emergency cases but has a high mortality rate. Early diagnosis is now possible with advanced imaging technologies that can result in the early start of anticoagulant therapy, preventing irreversible damage to the intestines. The development of direct thrombin and direct factor Xa inhibitors provides a safe anticoagulant treatment of AMI secondary to SMVT.


Mucinous cystadenoma occurs in 10–15% of all ovarian tumors. Diagnosis and treatment should be decided precisely as it has a chance to develop into pseudomyxoma peritonei (PMP). Management of PMP might be challenging especially when repeated surgery is needed.The first case, a 22-year-old lady with recurrent stomach enlargement for seven months. She had history of laparotomy surgery due to an ovarian tumor. Whole abdomen contrast CT scan showed a large cyst with mucinous fluid. We decided to do re-laparotomy and found a left ovarian cyst. Histological examination results confirm ovarian mucinous cystadenoma. The second case was, 55-year-old woman, with abdominal enlargement for six months. She had a history of laparotomy and chemotherapy due to pseudomyxoma peritonei. Post chemotherapy MRI showed persistent pseudomyxoma and two multilocular cysts from both adnexa. Debulking laparotomy was then conducted. We obtained 8 L of mucinous pseudomyxoma along with mucinous cyst from both ovaries. The final diagnosis concluded as a pseudomyxoma and we decide to close the follow-up of the patient.Pseudomyxoma is caused by the production of mucin originating from intra-abdominal organs. Open surgery should be prioritized when the mucinous cystadenoma is detected to do a complete peritoneum evaluation and avoid perioperatively ruptured mucinous neoplasm. Pseudomyxoma often needed repeated surgical treatment and may exhibit different surgical findings and different pathologies.Repeated surgery is logical and still no need for adjuvant chemotherapy in both cases. Accurate and precise diagnosis should be prioritized in order to prevent repeated surgery. •Accurate and precise diagnosis seems need to be prioritized in order to prevent repeated surgery.•Histopathological discrepancies need the expertise of the pathologists who specialize in gynecologic oncology.•Repeated surgery is logical and generally no need for adjuvant chemotherapy. Diagnosing mucinous cystadenoma should be decided precisely, regarding the treatment as a chance of becoming PMP 22.1A 22-year-old lady came with recurrent enlargement of the stomach for seven months. She had history of laparotomy surgery in 2015 due to an ovarian tumor. The histopathological result showed immature teratoma of the right ovary and endometriosis of the left ovary. In 2019, she had recurrent stomach enlargement and underwent surgery . Histopathological results of this surgery showed right ovarian mucinous cystadenoma, inflammation of the omentum and a normal appendix.Again in 2021, she came to our hospital with a third recurrent. The abdominal mass limited mobilization sized 30 cm in diameter. Laboratory findings found CEA 1.0 ng/ml, Ca 19–9934.6 U/ml, Ca- 125 24.9 U/ml. Whole abdomen contrast Computed Tomography (CT) scan showed a large cystic multilocular septated lesion with mucinous fluid and few solid components in the pelvic cavity and upper abdomen . We deci3 ascites, left ovarian cyst diameter of 40 cm. Fibrin adhesion with a rectosigmoid colon. Uterine was normal-sized and there was no more right adnexa (history of prior surgery). Left salpingo-oophorectomy was performed and the frozen section result was benign mucinous cystadenoma  and 5.4 × 5.1 × 5.8 cm (left) .Fig. 3CTThe patient was planned for HIPEC procedure, but due to not being feasible to manage, the patient was decided to change treated with FOLFOX regimen  and finally receive 5-fluorouracil (FUFA) and leucovorin according to availability of the national health insurance.After completing six courses chemotherapy, MRI was performed, and the results showed that accumulated fluid appeared heterogeneous after contrast, filling the entire intraperitoneal cavity from the upper to lower abdomen. Two multilocular cysts filled the pelvic cavity to the abdominal cavity measures approximately 14.6 × 21.8 × 30.5 cm (right) and 9.5 × 11.0 × 12.9 cm (left) .Fig. 4MRThe patient was consulted to Onco-Gynecology regarding an ovarian mass. Examination at the gynecological oncology distended abdomen with an abdominal girth of 100 cm. Inspeculo examination found a posterior vaginal bulging. Rectovaginal examination showed the mass filled the abdominal-pelvic cavity to fill the Douglas cavity. Laboratory examination showed CEA 117 ng/ml, CA-125 46 U/ml, and CA19-9 311 U/ml.The patient was planned for a debulking laparotomy and a total hysterectomy. On exploration, there was 8 L of mucinous pseudomyxoma obtained from the abdominal and pelvic cavities. Both mucinous ovarian cysts diameter were 15 cm with massive spreading mucinous implant until mesocolon, mesoilleum, subhepatic and subdiaphragmatic area. Surgical procedures were total abdominal hysterectomy, bilateral salpingo-oophorectomy, omentectomy, partial  peritonectomy, but not reach subdiaphragma stripping peritonectomy due to risk of diaphragma perforation. The abdominal cavity was then washed with 5% dextrose and 4 L of distilled water. A 32F drain catheter was installed for the preparation of intraperitoneal chemotherapy .Fig. 5A.As the previous discussion with internal medical-oncologist, surgical procedure performs pigtail drain catheter for HIPEC preparation procedure later after surgery. Histopathological results of surgical specimens showed mucinous pseudomyxoma of the fluid and the mucinous containing component of the ovarian cystadenoma. The final diagnosis concluded as a pseudomyxoma peritonei and decided no more treatment for HIPEC or chemotherapy and just closed follow-up .Fig. 6ImHER2 expression was found in borderline mucinous cystadenoma, but not found in PMP. It seems that positive HER2 protein was not detected in normal or benign tumor. Borderline tumors show HER2 positive staining, but mostly no overexpression In these cases, 34Pseudomyxoma can occur and caused by the production of mucin originating from intra-abdominal organs, including the ovaries, appendix, intestines, or peritoneal wall. First case illustrates that the final pathological result is a mucin cystadenoma that may cause ascites pseudomyxoma, but the ascites obtained do not contain mucin or pseudomyxoma elements to be included in the possibility of an early stage of pseudomyxoma. There was a different histopathology result from the first to the third examination in the first case. It revealed that the pathological examination findings at the first or subsequent surgery might be different because there are indeed differences in the components of the tissue structure in one part of the ovary, or it could be due to the transformation of cells from the ovary. Likewise, pathological examination with frozen section, it is possible that the results will be inaccurate compared to the results of paraffin block pathology. Such a condition was due to the accuracy of the frozen section, which significantly lower for mucinous ovarian tumors than other histological types. The causes of misdiagnosis in frozen section biopsy are mucinous ovarian tumor larger than other histological types, lower number of frozen section slides, experience or expertise of the pathologists who not specialized in gynecology oncology In the second case, despite having given a chemotherapy regimen on the first surgical biopsy findings that showed pseudomyxoma, it did not respond, even the mucin production was increasing. Further planning by the surgical team will be debulking and preparation for HIPEC chemotherapy, but the findings during the surgery showed that all components of ascites and right and left ovarian cysts contained mucin material and were not malignant. Based on the findings of the latest pathology results were pseudomyxoma, no malignant cells were found, it was decided that the therapy was only close follow-up without chemotherapy.We only did cytoreductive surgery based on our second case without HIPEC procedure. According to 5-year survival rates study conducted by Jarvinen et al., there was no difference between cytoreductive surgery alone compared to combination cytoreductive surgery and intraperitoneal chemotherapy Pseudomyxoma cases often receive repeated surgical treatment and may exhibit different surgical findings and different pathologies. However, in conclusion, both cases showed mucin cysts and pseudomyxoma which did not require chemotherapy 5Repeated surgery is logical and still no need for adjuvant chemotherapy in both cases. Accurate and precise diagnosis should be prioritized in order to prevent repeated surgery.5.1The third time of my stomach enlargement for seven months in 2021. My haid cycles were regular within the four last months. My doctor asks me whenever I had nausea, abdominal pain, and fever, but I felt none of it. I took Whole abdomen contrast Computed Tomography (CT) scan as a doctor's recommendation. And finally, the doctor decided to do re-surgery with informed consent that if my left ovary was not normal or don't have any function anymore, it may be decided to remove it. All of those considerations are based on the pathologist's examination in the middle of surgery. The final diagnosis of ovarian mucinous cystadenoma. And the medical team took a very hard decision to remove my left ovary, because it may always be a possibility of recurrence in the future. My family and I accept this hard decision, but we know that it will the best treatment for my condition.5.2The first symptom I felt was the enlargement of my stomach. At first, I just thought it was due to normal weight gain. However, as it turned out, my stomach grew rapidly in a relatively short period. After my first surgery in 2020 and chemotherapy, I felt a sense of relief, but soon the feeling of fullness in my stomach returned. In the end, I decided to be referred to a gynecologic oncology specialist. The discovery of 2 lumps from my ovaries and the accumulation of fluid in my abdominal cavity made the symptoms of shortness of breath appear. The 2nd operation was then conducted, and it went well. I felt that my abdominal circumference was much smaller, and the feeling of tightness in the abdominal cavity was reduced. I was then observed for four days post operative and then discharged.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.Not commissioned, externally peer-reviewed.This study was reviewed and approved by the Institutional Review Board and Ethical Committee Dr. Cipto Mangunkusumo, a national reference, and teaching hospital. Patient medical records were maintained under applicable medical ethical standards.This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.Gatot Purwoto.None declared.Gatot Purwoto: conceptualization, methodology, resources, supervision. Gatot Purwoto, Kelli Julianti, Nathaniel Herlambang, Ilham Utama Surya: writing-original draft preparation, investigation, visualization, writing-review and editing. Gatot Purwoto, Tricia Dewi Anggraeni: supervision, data curation, editing. Gatot Purwoto, Primariadewi Rustamadji, Kelli Julianti: visualization, analyzing, and interpretation data.The authors declare that we have no financial or personal relationship that may have inappropriately influenced us in writing this article.


This case report details a clinically rare presentation in which a middle-aged man was diagnosed clinically with a large irreducible inguinoscrotal hernia. However, intraoperatively, a large volume of old blood/clots was seen and aspirated, without a definite hernia being identified. Inguinal hernias remain a clinical diagnosis, and imaging is used only in equivocal cases. Owing to the number of differential diagnoses associated with groin swelling, careful clinical assessment is critical in differentiating between the various causes. Rupture of the inferior epigastric vessels was suspected, and although rare, it should be considered as a differential diagnosis as this may alter ongoing management. The inferior epigastric artery originates from the external iliac artery just above the inguinal ligament. It ascends superiorly and medially toward the umbilicus and supplies the muscles of the anterior abdominal wall. It forms an anastomosis with the superior epigastric artery and is accompanied by the inferior epigastric vein along its course .Owing to this anatomical location, these vessels can be injured and can lead to life-threatening haemorrhage. Iatrogenic injury from the insertion of lateral laparoscopic trocars has been reported in up to 2% of operative laparoscopies ,2. They Spontaneous inferior epigastric vessel rupture is associated with sudden-onset acute abdominal pain, swelling, and rectus sheath haematoma . TreatmeSpontaneous rupture of the vessels is a rarely reported phenomenon ,4 with lWe report on a rare clinical presentation in which a large collection of old blood was found masquerading as a large inguinoscrotal hernia. The source of the bleeding was suspected to have arisen from the inferior epigastric vessels.A 66-year-old Asian man presented to the surgical outpatient clinic with a 12-month history of worsening left-sided groin pain and swelling. His symptoms had recently begun to worsen with increasing discomfort, but despite this, he continued to work full time in a manual job. He denied any nausea or vomiting and there were no bowel or urinary symptoms. Furthermore, he was systemically well with no chest pain, dyspnoea, or dizziness.The patient’s medical history included type 2 diabetes and hypertension. He had no previous significant surgical history, and his daily medication included metformin, amlodipine, and ramipril. He had no known drug allergies and was not on any anti-coagulants. The patient was a non-smoker and denied alcohol intake.Physical examination revealed an overweight male with a soft, non-tender but distended abdomen. He had a gross irreducible left-sided inguinoscrotal swelling which was mildly tender on palpation. There was no bruising or overlying skin changes. Examination of the right groin was unremarkable. His baseline observations were entirely normal with haemodynamic stability. Urinalysis did not show any evidence of infection.The patient was diagnosed clinically with an irreducible large left inguinoscrotal hernia and counselled for surgery.An open approach was adopted, with an incision made in the groin crease. The regional anatomy proved difficult to identify, and no obvious hernial sac could be found. The decision was made to convert to a laparoscopic totally extraperitoneal (TEP) approach. A large collection of old blood and clots was found in the extraperitoneal plane tracking down to the scrotum Video . This waThe patient was followed up 7 days post-operatively in clinic and both Redivac drains with minimal haemoserous fluid were removed. A further review of the patient several weeks later found that he had recovered well with no signs of wound infection.Differential diagnoses of groin swelling include inguinal hernia, abscesses, haematomas, aneurysms, tumours, and lymphadenopathy. Additionally, in males, pathology arising from the testes, cord, and scrotal structures must also be considered.Careful history taking, physical examination, and the use of imaging modalities where appropriate can help to distinguish between these and guide further management. Imaging is seldom used in the diagnosis of inguinoscrotal hernias, especially in cases of high clinical suspicion, but may be employed in diagnostic uncertainty. The preferred mode of investigation is a computed tomography (CT) scan with contrast, especially in hernias likely to contain strangulated/incarcerated bowel. Alternatively, ultrasound scanning is a sensitive, specific, and contrast-free mode of imaging, but its quality remains operator-dependent.Inguinoscrotal hernias have a prevalence of 4% in males over the age of 45 years in the United Kingdom . PatientBy contrast, spontaneous haemorrhage secondary to ruptured inferior epigastric vessels leading to groin swelling and pain is a rarely reported presentation -8. It moIn this case report, imaging was precluded due to the clinical suspicion of inguinoscrotal hernia. The patient’s occupation in manual labour together with extended history, haemodynamic stability, the lack of an obvious trigger for vessel rupture, and the examination findings made a haemorrhagic cause for his symptoms unlikely. The unusual peri-operative findings coupled with this presentation are indeed a rare finding.Although the differential diagnoses of groin swellings are numerous, consideration should be given to the more uncommon presentations such as haematoma. Certainly, a history of localised abdominal trauma or the presence of shock should make clinicians suspicious of a haemorrhagic process. In cases where there is diagnostic uncertainty, imaging modalities can be utilised to aid assessment and help in further management.Management of such patients is usually dictated by the clinical presentation. In cases where there is haemodynamic stability, conservative management can be considered. Increasingly, interventional radiology offers an additional route of management with the option of embolisation. Ultimately, in cases where there is unstable haemorrhage, surgery with ligation of the offending vessels remains a viable option.


Osteochondromas, also known as exostoses, are the most common benign tumors of bone and can be classified into isolated and multiple osteochondromas. A great majority of osteochondromas is asymptomatic, painless, slow-growing mass, and incidentally found. However, osteochondromas occurring in adolescence or in adult patients can grow in size and become symptomatic as a result of mechanical irritation of the surrounding soft tissues or peripheral nerves, spinal cord compression, or vascular injury.We present a case of a 13-year-old girl with spontaneous hemothorax, the cause of which was identified by limited thoracotomy with the aid of video-assisted thoracic surgery to be bleeding from a diaphragmatic laceration incurred by a costal exostosis on the left sixth rib. Preoperative chest computed tomography (CT) depicted a bony projection arising from the rib and bloody effusion in the intrathoracic cavity, but was unable to discern the bleeding cause from the lung or the diaphragm. This case will highlight our awareness that costal exostosis possibly results in bloody pleural effusion. Meanwhile, English literatures about solitary costal exostosis associated with hemothorax were searched in PubMed and nineteen case reports were obtained. Combined our present case with available literature, a comprehensive understanding of this rare disease entity will further be strengthened.Injury to the diaphragm is the primary cause of hemothorax caused by costal osteochondroma, including the present case. Thoracic CT scan can help establish a diagnosis of preoperative diagnosis of costal osteochondroma. Surgical intervention should be considered for those patients with symptomatic osteochondroma of the rib. Combined with our case and literature, prophylactic surgical removal of intrathoracic exostosis should be advocated even in asymptomatic patients with the presentation of an inward bony spiculation. Osteochondromas represent the most common bone tumor accounting for 20–50% of all benign osseous tumors . They deOn August 12, 2021, a 13-year-old girl presented to our hospital with persistent left shoulder pain for more than 2 months, which was accompanied by left chest pain for 2 days. She denied the history of any familial diseases, recent chest trauma or having anticoagulant drugs. Physical examination revealed reduced respiratory movement and decreased breathing sounds in the left lung field. Percussion sounds of the left chest were solid. Besides, blood hemoglobin levels on admission was 108 g/L and clotting parameters were also normal. Chest CT demonstrated a left pleural effusion associated with passive atelectasis of the lung and a 2 cm long spear-shaped osseous proliferation originating from the visceral side of the left sixth rib  may be of some use in defining the extent of tumor spread locally. Compared with plain X-ray film, cross-sectional imaging offers a more accurate tool in the diagnosis of this condition. Recognition of the radiologic spectrum of osteochondroma and its variants usually allows prospective diagnosis and differentiation of the numerous potential complications, thus helping guide therapy and improving patient management . As descSurgical removal of osteochondromas is not usually indicated, especially in childhood. However, surgical resection is indicated for osteochondromas developing in adolescence after puberty or in adult patients with pain, increased size, mechanical complications and malignant transformation. Surgical management of thoracic osteochondroma, with excision for painful, symptomatic, malignant lesions or lesions adjudged to be at risk of intrathoracic complications, yields good outcomes in terms of symptom control, establishing histologic diagnosis, and prevention of thoracic complications . Cases oOsteochondromas are a very common entity, but rarely occur at the ribs. Osteochondroma of the rib may cause non-specific, occasionally serious complications for mechanical frictions or mass effect. Thoracic CT scan can help establish a diagnosis. Surgical intervention should be considered for those patients with symptomatic osteochondroma of the rib. Combined with our case and literature, prophylactic surgical removal of intrathoracic exostosis should be advocated even in asymptomatic patients with the presentation of an inward bony spiculation.


Mesenteric lipoma is a rare entity. It can be asymptomatic or revealed by unspecific clinical symptoms. Complete resection of the lipoma is often proposed to prevent complications. We report a case of mesenteric lipoma revealed by chronic abdominal pain, and we performed a literature review regarding this rare condition. Mesenteric localization of lipoma is very rare. It may be suspected in patients presenting with chronic abdominal pain. Complete resection of the tumor is indicated for symptomatic or complicated mesenteric lipoma. It commonly occurs in the cephalic part of the body.2A 22‐year‐old man with no medical history presented to the hospital with abdominal pain located in the right iliac fossa. The pain began 2 years ago with aparoxysmal evolution, but it has worsened over the last 6 months. The patient also complained of chronic abdominal discomfort. There were no signs of bowel obstruction. Physical examination found a tenderness of the right iliac fossa without a palpable mass. Biology tests were normal including C‐reactive protein. We performed an abdominal ultrasound, which showed a well‐defined, homogenous mass of the right iliac fossa of 10 × 8 cm. The abdominal computed tomography (CT) scan showed a well‐defined mass in the mesentery measuring 12 × 9 cm with fat density surrounded by a thin capsule without any septum or calcification.  is also very performant to describe in detail the characteristics of the mass. It shows a hypointensity on T1‐weighted and T2‐weighted images without modification of the signal after injection of gadolinium.Therapeutic management of mesenteric lipoma is not consensual, and it is based on the experience of the different teams. A small lipoma in a asymptomatic patient can be respected.The recurrence rate of all lipomas after surgery is less than 5% and is usually due to incomplete excision.4Although mesenteric lipoma is a rare condition reported in the literature, it should be considered in the differential diagnosis of unexplained chronic abdominal pain. Contrast‐enhanced CT scan is the gold standard diagnosis procedure. Treatment is based on complete surgical resection with or without the affected intestinal loop, particularly, in case of symptomatic or large lipoma to prevent the risk of complications.5This report describes a rare case of mesenteric lipoma revealed by chronic abdominal pain in 22‐year‐old man. The CT scan images were most consistent with a lipoma and showed no signs of malignancy. Complete excision of the lesion was performed with a clear plan of division between the tumor and the ileal mesentery. Histopathologic examination confirmed the diagnosis of benign lipoma. The postoperative period was uneventful, and no recurrence has been diagnosed. In conclusion, mesenteric lipoma should be considered in the differential diagnosis of unexplained chronic abdominal pain.None declared.Kais Fourati, Amine Zouari, Rami Zouari, and Jawhar Bradai involved in patient management. Aberahmen masmoudi and Jawhar Bradai collected the data. Kais Fourati, Abderahmen Masmoudi, Amine Zouari, and Rami Zouari drafted the manuscript. Najmeddine Hentati and Salah Boujelbene involved in manuscript revision and supervision. All authors read and approved the final manuscript.Personal data have been respected.Written informed consent was obtained from the patient to publish this report in accordance with the journal's patient consent policy.


Inflammatory granulomas often develop in surgical scars due to the presence of foreign bodies, such as sutures. These granulomas are called Schloffer’s tumors. Here, we report a case of heterotopic ossification(HO) in an appendectomy scar that formed an inflammatory granuloma following HO infection. A 90-year-old woman was referred to our hospital with a chief complaint of a painful mass in the right lower quadrant of her abdomen. She had a history of acute appendicitis, for which she underwent an appendectomy approximately 70 years previously. Imaging studies demonstrated a tumor containing a linear-shaped agent located in the abdominal wall under the surgical scar where the appendectomy was performed. She was then diagnosed with Schloffer’s tumor, for which she underwent surgical resection. However, histopathological examination revealed that the tumor was a fibrous connective tissue mass with a lamellar bone inside. Inflammatory granulomas often develop in surgical scars due to the presence of foreign bodies, such as sutures. These granulomas are called Schloffer’s tumor . EndogenA 90-year-old woman was referred to our hospital complaining of a painful mass located in the right lower quadrant of her abdomen. She had a history of acute appendicitis, for which she underwent an appendectomy approximately 70 years prior to the current consultation. She first noticed the pain 6 months prior to consultation. In the interim, the pain gradually worsened. Upon physical examination, a palpable tumor was present below the appendectomy scar. Yellowish pus was removed from the skin over the tumor . ComputeUnder the preoperative diagnosis of foreign body granuloma, also known as Schloffer’s tumors, which contained abscess, the patient underwent surgical resection of the tumor. The tumor was 60 × 40 × 40 mm, slightly hard and capsulized. It also had a stiff and wiry body .Histopathological examination of the tumor revealed fibrous connective tissue with abscesses and bacteria. Diffuse infiltration of inflammatory cells, such as neutrophils and histiocytes, was observed . The wirTumors that grow on surgical scars are often diagnosed as Schloffer’s tumor. However, in this case, heterotopic bone tissue, instead of a foreign body, was found in the tumor. Although HO is not common, it can be one of the causes of granuloma development in surgical scars. We speculate that HO developed in the appendectomy scar 70 years after the operation. Thereafter, the infected HO tissue formed a granuloma and an abscess. To the best of our knowledge, there have been no reports of HO accompanied by granuloma generation.et al. first reported HO in an abdominal scar in 1901 [HO is the formation of bone outside the skeletal system, which can occur in all types of soft tissues, such as skin, scars, subcutaneous fat, muscle and mesenterial tissue . Askanaz in 1901 . In a re in 1901 . In JapaThe pathogenesis of HO has not yet been fully elucidated. Nevertheless, several theories have been proposed. Some studies suggested that vertical incisions in the proximity of the xiphoid process or pubic symphysis inoculated periosteal or perichondrial cells. This ‘seeding’ of the surgical wound with activated osteoprogenitor cells could therefore induce formation of heterotopic bone . Other set al. reported the role of BMPs in heterotopic bone formation, and suggested that inflammation and activation of BMP signaling are necessary for the development of HO [Recent studies proposed that bone morphogenetic proteins (BMPs), which were involved in the regulation of bone induction, maintenance and repair, played critical roles in osteogenesis and chondrogenesis . Yu et ant of HO .In our case, HO developed in the right lower quadrant of the abdomen.Since there was no continuation of bone tissue nor periosteum, we assumed that immature pluripotent mesenchymal cells differentiated into bony tissue in the surgical scar. We assumed that surgical invasion, BMPs and inflammation-induced cell differentiation into HO. Although no studies reported a case of an infected HO, bacterial infection of HO tissue seemed to occur through the bloodstream from other sites. Some reports showed that acupuncture causes ectopic infection, thereby resulting in piriformis muscle abscess, trapezius muscle abscess and necrotizing soft-tissue infection , 11. OurWe initially diagnosed the tumor as Schloffer’s tumor. They are caused by infection of foreign bodies, such as sutures left in the abdominal wall, which then generates granulomas. On the other hand, few patients were diagnosed with Schloffer’s tumor because it existed in the surgical scar, although no foreign body was detected. We concluded that our case was not Schloffer’s tumor because the tumor was derived from an intrinsic lamellar bone.We performed the surgery for two indications. The first indication was the symptomatic presentation of the patient. The second reason was to arrive at as definitive diagnosis.We reported a very rare case of infected HO that grew on an appendectomy scar and resulted in an inflammatory granuloma and abscess 70 years after the surgery.C.H. and Y.T. substantially contributed to the conception and design of the case report. They also drafted the manuscript. K.M. acquired the pathological data. K.K., M.M., R.T., K.S., T.H., T.D., A.T., K.G., M.M., K.T. and M.H. contributed in drafting the manuscript. T.K. critically revised the manuscript. All authors have read and approved the final manuscript.


Desmoplastic infantile ganglioglioma (DIG) has a favorable prognosis and is classified as a benign infantile brain tumor. The DIG is more common in children under 2 years of age than in other age groups. This report introduces a 5.5 month-old infant who was referred with infantile spasms and diagnosed with a brain tumor. Brain magnetic resonance imaging showed a large heterogeneous mass in the right hemisphere with shifting to the other side. The patient underwent surgery. The extra-axial mass was completely resected, and the diagnosis of DIG grade I was confirmed by pathology. After one year, patient development was normal, and the seizures did not recur. In addition, the general condition was good. With a brief review and search in the literature, 13 case reports were identified 9 of which were male cases. The mean age of initial manifestation to final tumor diagnosis was 4 months. Out of 13 patients, 8 cases were reported with the mass origin in the right hemisphere. The most commonly observed tumors were glioma (n=4) and hypothalamic hamartoma (n=3). Except for three patients who died, the remaining had a complete recovery after tumor removal with a seizure-free interval at follow-up. Epileptic spasms are regarded as a known type of seizure occurring with an incidence of 25 per 100,000 live births in the United States and Western Europe. An underlying cause can be determined in approximately 75% of patients. The etiologic classification in infantile spasms has been recommended as genetic, structural/metabolic, or unknown causes. Structural causes, such as brain tumors, are among uncommon etiologic factors that probably clinically present with infantile spasms ( Desmoplastic infantile ganglioglioma (DIG) is a rare infantile brain tumor that rarely appears with infantile spasms. The DIG  is primarily observed in infants and young children, with a male predominance. Patients with DIGs typically present in infancy with macrocephaly or partial complex seizures. The median reported age of presentation is about 5 months. The involvement of multiple lobes is common with a predilection for the frontal and parietal lobes .  Vandenberg was the first who described DIG in 1987 . The DIG Most of the present studies have reported that infantile spasms have appeared with central nervous system diseases; however, the occurrence of infantile spasms with brain tumors is not common . In thisThe patient, a 5.5-month-old infantile male, was born after an uneventful full-term pregnancy and a natural vaginal delivery. His birth weight and head circumference were 3200 g and 34 cm, respectively. The family history included a father with thalassemia major and a cousin treated for hydrocephalus. He was referred to the hospital with recurrent epileptic spasm attacks, mainly while waking up, lasting less than 20 seconds. His attacks started 2 weeks before the referral. The spasm was extension type with nystagmoid eye movements. During the physical examination, he was conscious and had attention to his surroundings. He did fix and follow. On evolutionary examination, he comfortably held his neck and rolled over. Eyes function and limb strength were normal. Reflexes were asymmetric with exaggerated reflexes in the left lower limb and mild left hemiparesis. Moreover, no skin lesions were observed.An electroencephalogram (EEG) showed a burst suppression pattern, indicating modified hypsarrhythmia and disorganized background with mild asymmetry between the two hemispheres  and 2. TThe brain MRI of the patient exhibited large heterogeneous multilobulated and cystic masses in the right temporoparietal region with shifting to the left . After iThe patient underwent surgery . The extOne of the most critical seizures in infancy is an infantile spasm. Infantile colic is one of the differential diagnoses of infantile spasms. Additionally, infantile spasms will have a worse prognosis if it is not diagnosed early and treated. The causes of infantile spasms are symptomatic (80%), cryptogenic, and idiopathic. Common symptomatic causes include neonatal ischemic hypoxic encephalopathy and tuberous sclerosis, which do not have a favorable prognosis. Less common causes include genetic and acquired structural disorders. Brain tumors are regarded as a structural and acquired disorder clinically presenting as infantile spasms .According to the Google search for the words brain tumor and infantile spasms, 13 patients have been reported. The onset of symptoms varied from 1 to 14 months, and the mean age of onset of symptoms was 4 months, which is approximately consistent with the age of onset of symptoms in the present patient  -13. AccoOf 13 patients, 8 patients were reported with the origin of the mass in the right hemisphere. The origin of the mass in three patients was the left hemisphere , 9, 10. In addition, in the present case, the final pathology of DIG was reported. Except for one patient, all patients had hypsarrhythmia and modified hypsarrhythmia in the EEG. Moreover, as a common electrophysiologic finding, modified hypsarrhythmia was also reported in this case  and 4. Except for three patients who died (the first case from glioma grade III , the secThis study received no specific grant from any funding agency in the public, commercial, or not-for-profit sectors. All authors had full access to the data, contributed to the study, approved the final version of the manuscript, and took responsibility for its accuracy and integrity. The authors have no conflict of interest in preparing and submitting this report.


To describe demographic, clinical, diagnostic and therapeutic aspects of pediatric patients with benign adipocytic tumors admitted to a high complexity teaching hospital from 2007 to 2021.Retrospective observational descriptive study. Patient information was retrieved from clinical records. A descriptive analysis was carried out for qualitative data and frequencies were calculated for quantitative data.76 patients were included with a mean age of 7.5 years old where 60.5% were boys. The main symptom was a mass (73.7%) mostly found in the lower limbs (23.6%). Congenital birth defects were identified in 48.6% of the cases. Preoperative imaging was available in 78.9% of the patients allowing characterization of lesions or differential diagnosis. The therapeutic goal was resection with negative margins, which was feasible in all cases except for one case. The histopathological diagnosis was lipoma in 68.4% of the cases followed by lipoblastoma in 13.1%. The mean follow-up period was 17.9 months. 79.7% of the patients were asymptomatic at their last out-patient visit.Benign adipocytic tumors constitute a wide spectrum of lesions, which involve diverse anatomic segments from the neural axis to the inguinoscrotal region. The present work contributes to the general understanding of the clinical presentation and differential diagnosis for these infrequent neoplasms. Adipocytic neoplasms account for less than 10% of soft tissue tumors in the pediatric population. They are histologically distinct from adult tumors and can be associated with genetic syndromes . There iDifferential diagnosis of soft tissue masses in pediatric patients requires knowledge of the patient’s age, time of onset, location, variations in size, cutaneous changes, and comorbid conditions. Taking this into account, most adipocytic tumors are present as superficial, usually painless, masses located in the limbs. Ultrasound (US) is the first-line test for diagnostic evaluation given its availability, low cost and lack of ionizing radiation and exposure to contrast agents . HoweverOn microscopic examination benign lipomatous tumors resemble white adipose tissue with variable differentiation grades . The theA retrospective descriptive observational study was designed with the approval of the Institutional Ethics Committee (Act No. 45 382–21). Data were retrieved from the Fundación Hospital Pediátrico la Misericordia pathology laboratory database, Bogotá D.C., Colombia. 107 records with histopathological diagnosis of benign lipomatous tumors were identified between January 2007 and July 2021. Cases with findings that were compatible with variants of “lipoma” and “lipoblastoma” were included for a total of 76 patients. 31 cases were excluded. 8 of them were duplicate registries from a same patient. 23 were referral cases from a regional institution to our Pediatric Pathology Laboratory, therefore clinical records were unretrievable.Demographic and clinical data including age, sex, comorbidities, main complaint, associated symptoms, mass location, diagnostic imaging, surgical approach, and histopathological diagnosis were obtained by reviewing medical records. A follow-up period was accurately recorded for 70 cases and was defined as the time in months beginning from diagnosis until the last out-patient visit. Due to the nature of the study, no statistical analysis was performed. Nonetheless, descriptive statistics including mean, median and range were calculated for quantitative data and frequencies expressed as percentages for qualitative data.n = 56, 73.7%), followed by pain , deformity  and others such as cough, gait instability, joint edema, dysraphism and myoclonic crisis , where 60.5% of the population were male and 39.4% were female, with a male:female ratio of 1.5:1. The most frequent symptom was a mass , followed by dorsolumbar soft tissues , inguinal region , upper limbs , spinal canal , neck , chest wall  and retroperitoneal location compromising renal parenchyma . Other less frequent locations were the gluteal region , perineum , abdominal wall , frontal region , scrotum , oral cavity  and peritoneal cavity compromising the omentum .The most frequent findings were observed in the lower limbs , epileptic syndromes mostly associated with tuberous sclerosis (13.8%) and anorectal malformations (5.5%) were the most frequently described. Two procedures were carried out on a patient with lipoblastomatosis who was previously diagnosed with Proteus syndrome.Pre-procedural imaging was performed in 78.9% of the population studied. The first diagnostic test was an US, which was performed in 32.8% of the cases, 21% had a CT scan and 51.3% had a MR imaging. On 38.9% of the cases two imaging tests were performed, while only two cases had the three tests performed (3.3%). Most lesions evaluated by sonography described solid, hypoechogenic masses, with well-defined lobulated margins sparing muscular structures, some of which displayed intralesional longitudinal echogenic images and absence of flow on Doppler US mode, except for vascularized masses, such as angiolipomas . On microscopic evaluation these lesions were characterized by the presence of mature adipocytes arranged in lobules separated by mature fibro-connective tissue strands, with myxoid stromal foci and capillary vascular proliferation . On microscopic evaluation these lesions were characterized by the presence of cells with variable differentiation grades, including prelipoblasts, lipoblasts, preadipocytes, multivacuolated adipocytes and mature adipocytes , fibrolipoma  and diffuse lesions including lipomatosis  and lipoblastomatosis .Tumor size varied between 0.5 and 20 cm . In all age groups the main histopathological diagnosis was lipoma (tes Fig. . The remOur cohort agreed with previously published studies regarding the mean age of adipocytic tumor presentation , as wellDepending on their location, adipocytic tumors can be associated with other manifestations such as dysraphism , abdominOne patient from our cohort underwent multiple resections for lipoblastomatosis associated with Proteus syndrome. This condition is characterized by diffuse soft tissue overgrowth and can be associated with other disorders, such as tuberous sclerosis . Other dFor superficial lesions US was the diagnostic tool of choice in contrast with cross sectional imaging studies, which aimed to characterize masses with compromise of deep tissue compartments and body cavities. In most cases diagnosis was accurately determined by imaging; however, some reports required differential diagnoses, such as lymphatic malformations or alternative neoplasms, which can also contain adipose tissue. Furthermore, in this series an important number of lesions were identified as an intraoperative incidental finding in unrelated pathologies, such as testicular torsion or spermatic cord hydrocele. Even though CT imaging has limited applications for soft tissue evaluation, such as lipomatous tumor diagnosis , 21% of In this series, most complications were related to factors associated with the resection site, unlike other cohorts which did not describe complications. In the present study reported recurrence rate was low, as had been described in the literature for lipomas, corresponding to less than 5% of the cases . HoweverThe mean tumor size was variable when compared to previous studies . HoweverThe present study aimed to report the local epidemiology of benign adipocytic tumors through the description of one of the largest case series to date centered on the clinical presentation, comorbid conditions, histopathological diagnosis, and complications associated with surgical management. The focus on benign pathology was motivated by the increasing diagnosis of lesions compatible with lipoblastoma in our setting. The results of this study support a higher incidence of lipomas in all age groups, unlike previously published literature that reports a higher frequency of lipoblastomas in the group of children under three years of age .One of its limitations was the follow-up period of less than 5 years, which is the time-period usually recommended by some authors to increase the probability of identifying late recurrences . AnotherFor instance, 12q14–15 and 6p21 loci alterations have been shown to result in overexpression of HMGA2 and HMGA1 in pediatric lipomas. Similarly, it has been possible to characterize the overexpression of PLAG1 related to 8q11 locus alterations in lipoblastomas. The identification of these molecular patterns, as well as the diagnosis of other associated syndromes could have important therapeutic and prognostic implications in patients with adipocytic neoplasms in the future [Local epidemiology of benign adipocytic tumors in children in our setting is similar to that previously reported in the literature; yet we identified a lower proportion of lipoblastomas. Additionally, to properly identify late recurrences a longer follow-up period is needed. Furthermore, tumor molecular characterization is relevant for establishing accurate diagnosis of atypical disease, unfortunately this technology is not readily available for routine use in the local practice Table .Table 1D


Myxofibrosarcoma (MFS) is a subtype of soft tissue sarcoma characterized by diffuse infiltration patterns. Myxofibrosarcoma arises often in extremities. Its occurrence in the abdominal wall is extremely rare.Herein, we present here a case of high-grade MFS of the abdominal wall discovered in a 58-year-old woman complaining of an abdominal mass.This report illustrates the case of a female who presented a mass in the right lower quadrant of the abdomen. Abdominal computed tomography (CT) scan revealed a well-circumcised, heterogeneous soft tissue mass. We performed a wide margin excision of the mass. Histology concluded in myxofibrosarcoma of the abdominal wall. Adjuvant radiotherapy was performed.We reported successful surgical treatment for myxofibrosarcoma of the abdominal wall. To our knowledge, this is the second report in English literature.MFS is a subtype of soft tissue sarcoma with a locally infiltrative behavior.To ensure the best curative treatment, It is important to excise the tumor with wide margins.Knowing that MFS has a propensity for local recurrence (16 to 57 %), adjuvant radiotherapy has emerged as an efficient treatment for improving local control.The role of chemotherapy is controversial and has not shown effects on survival.Myxofibrosarcoma is a connective tissue neoplasm. Its occurrence in the abdominal wall is extremely rare.Surgical treatment with large negative margins is the cornerstone of the treatment. Adjuvant radiotherapy is essential in preventing local recurrences. •Myxofibrosarcoma of the abdominal wall is an extremely rare situation.•Surgical treatment is the cornerstone of the treatment.•Adjuvant radiotherapy is an option to prevent relapses. It represents 5 % of soft tissue sarcomas MFS presents a significant propensity for local recurrence following resection (50 %) Herein, we present here a case of high-grade MFS of the abdominal wall discovered in a 58-year-old woman complaining of an abdominal mass.This work has been reported in line with the SCARE 2020 criteria 2A 58-year-old female, with no comorbidities, complained of a fast-growing abdominal mass evolving since one year. She had no other symptoms. She had no family history of cancer.On examination, the patient had a tender, non-reducible, non-mobile mass in the right lower quadrant of the abdomen.Laboratory studies revealed hemoglobin of 11.2 g/dl, platelet count of 230,000/μl, and prothrombin time of 97 %. Renal and hepatic functions were normal.Abdominal computed tomography (CT) scan revealed a well-circumcised, heterogeneous soft tissue mass measuring 13 × 10 × 8 cm of the abdominal wall , Fig. 2.The case was discussed in a multidisciplinary meeting. We considered performing a needle biopsy of the mass or performing surgery directly. We opted for the surgical treatment knowing that the mass was symptomatic and even if there was no malignancy, the mass had to be resected to relieve symptoms.Surgery was planned and was performed by a 15-year experience surgeon.We considered the diagnoses of a desmoid tumor and leiomyosarcoma of the abdominal wall.We performed an elective incision. We resected the mass and the adjacent skin, dermis, and subcutaneous tissue with a 1 cm margin . ReconstHistopathology showed a high-grade spindle cell tumor with scattered nuclear pleomorphism in a prominent fibromyxoid stroma . StaininThe postoperative course was uneventful with a follow-up of two months. Adjuvant radiotherapy was performed to prevent relapses.3We reported successful surgical treatment for myxofibrosarcoma of the abdominal wall. To our knowledge, this is the second report in English literature. Adjuvant radiotherapy was performed to prevent relapses.The main weaknesses of our work are the short follow-up period of only two months and the non-performance of MRI.MFS is a subtype of soft tissue sarcoma with a locally infiltrative behavior. It affects generally older patients. Men are affected more frequently than women The extremities are the most prevalent locations of presentation of MFS (77 %). They can also be seen on the trunk (12 %), and the head and neck area (3 %). MFS is rare in the abdomen, retroperitoneum, skin, breast, and chest wall It usually manifests as a slowly growing, painless mass. On computed tomography (CT), MFS present as a heterogeneous soft tissue mass. MRI is the diagnostic modality of choice. It shows a low to intermediate signal on T1-weighted MRI. On T2-weighted MRI, the solid and myxoid components show high signal intensity, with the myxoid component showing higher signal intensity similar to fluids Needle biopsy of the tumor is a considerable option We considered differentials such as Leiomyoma, Leiomyosarcoma, extra gastrointestinal stromal tumor (E-GIST), and liposarcoma Intermediate and high-grade sarcoma present a high risk of recurrence (50 %) and distant metastasis. To ensure the best curative treatment, It is important to excise the tumor with wide margins To date, there is no consensus on the width of surrounding normal tissue that should be excised Overall survival for patients having localized MFS varies from 61 to 77 % at 5-year The role of chemotherapy is controversial and has not shown effects on survival Preoperative radiotherapy can be considered in presence of tumor invasion of an important adjacent structure permitting down-sizing the tumor before surgery. This permits to avoid leaving positive resection margin.Further prospective cohort studies with a larger sample size could aid in assessing preoperative and postoperative radiotherapy in preventing local recurrence.In summary, we reported a case of MFS of the abdominal wall treated with surgical excision and adjuvant radiotherapy.4Myxofibrosarcoma is a connective tissue neoplasm. Its occurrence in the abdominal wall is extremely rare.Surgical treatment with large negative margins is the cornerstone of the treatment. Adjuvant radiotherapy is essential in preventing local recurrences.This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.Not required.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.Hazem Beji.Mahdi Bouassida.Not commissioned, externally peer-reviewed.N/A.Hazem Beji and Mahdi Bouassida did the conception and design of the work, the data collection, and the data analysis and interpretation.Mohamed Fadhel Chtourou and Slim Zribi did the critical revision of the article.Mohamed Mongi Mighri and Hassen Touinsi did the final approval of the version to be published.No conflicts of interest.


They are a subset of primary extradural meningiomas (PEM). In this study, we present a peculiar case with a systematic literature review and propose a new classification considering the limitations of previous classification systems. (2) Materials and Methods: Using a systematic search protocol in Google Scholar, PubMed, and Scopus databases, we extracted all case studies on PIOM published from inception to December 2020. A 46-year-old female patient form Dhaka, Bangladesh, was also described. The search protocol was performed according to the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) guidelines. (3) Results: Here, we present a 46-year-old female patient with PIOM who successfully underwent bifrontal craniotomy and gross total removal (GTR) of the tumor. At 6-month follow-up, no tumor recurrence was shown. Including our new case, 55 total cases from 47 articles were included in the analysis. PIOMs were in closer frequency among males (56.4%) and females (43.6%). The most common tumor location was the frontal and parietal calvarium, most commonly in the frontal bone (29.1%). Surgical resection was the predominant modality of treatment (87.3%); only 1.8% of patients were treated with radiotherapy, and 5.4% received a combination of surgery and radiotherapy. Gross total resection (GTR) was achieved in 80% of cases. Extracranial extension was reported in 41.8% of cases, dural invasion in 47.3%, and recurrence in 7.3%. Whole-body 68 Ga-DOTATOC PET/CT has also been reported as a useful tool both for differential diagnosis, radiotherapy contouring, and follow-up. Current treatments such as hydroxyurea and bevacizumab have variable success rates. We have also suggested a new classification which would provide a simple common ground for further research in this field. (4) Conclusions: Surgical resection, especially GTR, is the treatment of choice for PIOM, with a high GTR rate and low risk of complications and mortality. More research is needed on the differential diagnosis and specific treatment of PIOM.  Meningiomas are typically slow-growing tumors that arise from arachnoid cap cells . MeningiWe searched Google Scholar, PubMed, and Scopus databases for the selection of peer-reviewed published articles for our systematic review with appropriate mesh terms. Only case reports and case series of PIOM were found. Therefore, during the selection procedure, we screened published case reports and case series from the inception to December 2020 following the search criteria. We restricted the screening language to only English. The search terms included “primary intraosseous meningioma”, “primary intraosseous osteolytic meningioma”, and “PIOM” to incorporate all potential articles in our analysis. The Mendeley citation manager was used for the management of the articles collected through our systematic search . The stuTo meet the objectives of our study, we included all available case reports and case series regarding PIOM involving the skull vault and base and reviewed them meticulously. Papers lacking necessary information regarding demographic characteristics, clinical presentation, diagnostic modalities, treatment, histopathology, and outcome were excluded.The information from selected research articles were recorded in Microsoft Excel 2013. We further reviewed the articles for missing information and checked for consistency. Data analyses were conducted by IBM SPSS (version-23) statistical package software.  A 46-year-old female patient was admitted to the Neurosurgery Outpatient Department of Ibrahim Cardiac Hospital and Research Institute, Dhaka, Bangladesh, in 2019, complaining of a large subcutaneous mass in the frontal area. She first noticed a small, non-tender, hard lump in the mentioned area 8 years ago. The lesion increased very slowly over time. Two years ago, she presented a papillary carcinoma of thyroid and underwent total thyroidectomy. Due to the presence of the lesion, there was clinical suspicion of skull metastasis. As the patient denied any neurosurgical intervention, she was advised to receive whole-brain radiotherapy. After completion of radiotherapy, she noticed rapid enlargement of swelling, along with headaches. For the past 8 months, due to additional changes in her personality and behavior, she had an MRI of her brain and was referred to our department for further evaluation and management.Local examination of the mass demonstrated bony, hard, mildly tender swelling of 6 cm × 4 cm × 3 cm in the frontal region. The mass had ill-defined margins with an irregular surface, fixed with overlying skin as well as underlying structures. There were no palpable lymph nodes and no swelling elsewhere in the body. Metastatic work-up was negative.A plain X-ray of the skull showed an expansile lytic lesion having internal septations located in the frontal bone, with a bulging of overlying soft tissue shadow. There was no abnormal calcification. Vascular markings appeared to be normal; all the features were suggestive of metastasis. For better delineation of the pathology, an MRV was also performed. There was an irregular, lobulated extra-axial T1WI iso to hypointense and T2WI heterogeneously hyperintense mass measuring about 7.9 cm × 7.6 cm × 7.4 cm noted in both frontal regions . Mass efThe tumor was exposed through a bicoronal incision and subgaleal dissection. The mass presented diffuse infiltration of the subcutaneous tissue. After meticulous dissection, the flap was retracted antero-inferiorly . A bifroThe patient presented an uneventful recovery. There was no onset of new neurological deficits during follow-up. Post-operative head CT scan documented a complete tumor removal . She wasSections made from the submitted specimen show a meningothelial meningioma with whorl formation. The cells were epithelioid in shape, having oval nuclei. No mitosis or necrosis were seen .We presented a 46-year-old female patient treated for PIOM. Her follow-up head CT scan showed GTR, and the patient was clinically stable at 6 months. Including our new case, a total of 55 cases from 47 articles were considered for the analysis. The mean age of the study participants was 55.38 (range 6–84 years); 31 out of 55 (56.4%) were males and 24 out of 55 (43.5%) were females. The most common tumor location was the frontal and parietal calvarium, with the frontal bone being the most common occurrence (29.1%) of the cases, the parietal bone in 23.6%, and a combination of the frontoparietal bone in 10.9% of the cases. The most common symptom was a visible mass lesion, which occurred in 52.7% of the patients, and it was typically a growing mass. Surgical resection alone was the predominant modality of treatment, occurring in 87.3% of the cases. Only 1.8% of patients were treated with radiation alone, and 5.4% received a combination of surgery and radiation. Gross total resection was achieved in 80% of cases. The mean post-operative follow-up interval was 15 months. Extracranial extension was reported in 41.8% of cases and dural invasion was reported in 47.3% of cases. We categorized the PIOMs according to the histopathology following the WHO categories type I (74.5%), type II (16.4%), and type III (9.1%). Recurrence was reported in 7.3% of the patients.PIOM is a term used to describe a subset of primary extradural meningioma that arise in bone, when no dural attachment is present. They can present either as an osteoblastic lesion or an osteolytic lesion . The terThere are scant literature addressing the mechanism of osteolysis in PIOM. In 2007, Sade et al. showed integrin-mediated adhesion of osteoclasts to the bone matrix in the case of skull base meningioma, which promotes degradation of bone collagen by releasing lysosomal enzymes (ITG B1) . MoreoveAlthough meningiomas are the most common extra-axial tumor in adults, intraosseous meningiomas are rare tumors that originate in the skull, accounting for 1–2% of all meningiomas . The majIntraosseous meningiomas usually occur in both males and females with the same frequency or with a slight predominance among females . HoweverAccording to the literature, hyperostosis is present in 59% of PIOM imaging evidence, osteolytic changes in the surrounding bone appear in 32% of cases, and mixed features of osteolysis-hyperostosis are reported in 6% of cases . The bonA review conducted by Lang et al. identified dural involvement in 60% (CT or MR imaging) of PIM patients. On visual inspection after craniotomy, the dura appeared normal in 40% of the cases . AccordiSurgical resection (GTR) is the major treatment of choice for primary intraosseous meningiomas, with low risk of complications and mortality reported. When feasible, wide en bloc resection including 1 cm negative margins is recommended in high-grade meningioma . In the In our analysis, recurrence was noted in 7.3% of cases, which is lower than another previously conducted study, where recurrence was noted in 22% of cases of benign PEMs . On the The classification by Lang et al. was simple and useful from a topographical point of view, but it has some limitations . Some tuThis study revealed a new case of PIOM in Bangladesh which successfully underwent bifrontal craniotomy and gross total removal (GTR). Based on our analysis, we recommend complete resection as the treatment of choice for these PIOMs. Serial follow-up to confirm recurrence or progression should be conducted after the surgery. 68 Ga-DOTATOC PET/CT is a useful tool for differential diagnosis, RT contouring, and follow-up. The study also revealed a new classification which would assist researchers and clinicians in further research in this field and in decision making. More research is required on the mechanism of osteolysis, management strategies, and specific treatment.


Lower urinary tract symptoms are very common in elderly women, and transvaginal delivery and multiple deliveries have been confirmed to be risk factors. Transvaginal delivery and multiple deliveries may lead to an increase in pubic symphysis degeneration.A 79-year-old woman consulted a urologist because of worsening lower urinary tract symptoms such as frequent urination and urodynia. Color ultrasound and cystoscopy suggested the possibility of a bladder mass. A lump on the anterior wall of the bladder was observed although the surface mucosa was normal. Physical examination showed obvious tenderness in the posterior area of the pubic symphysis. Further urological computed tomography (CT) and pelvic magnetic resonance imaging (MRI) showed a nodular bony protuberance in the posterior part of the pubic symphysis, which was more obvious than before, with compression changes near the anterior wall of bladder. Open pelvic surgery showed that nodular bone tissue originating from the pubic symphysis significantly oppressed the anterior wall of the bladder behind the pubic symphysis. After resection of the nodule, the lower urinary tract symptoms were relieved significantly.Pubic symphysis degeneration caused by transvaginal delivery may be an important cause of lower urinary tract symptoms in women. Pelvic CT or MRI is necessary to diagnosis this condition.The online version contains supplementary material available at 10.1186/s12894-022-01052-1. Lower urinary tract symptoms are important factors that affect the quality-of-life of elderly women. Urinary tract infections, an overactive bladder, and relaxation of pelvic floor muscles are common causes of lower urinary tract symptoms . HoweverTwo years ago the patient, a 79-year-old woman, had complained of frequent urination and urodynia for which there was no obvious cause. The painful urination resulted in continuous severe pain behind the pubic bone after urination, forcing the patient to tilt forward with flexion and not dare to straighten-up. The pain was relieved gradually after about 15 min, but reoccurred at the next urination. Bladder color ultrasound revealed a bladder mass, but there was no gross hematuria. One year ago, the patient was treated in our hospital, with a urinary CT and pelvic MRI identifying an anterior calcified bladder nodule, adjacent to the pubic symphysis. The nodule was considered to be a lesion occupying the pelvic space, although gynecological examination showed no abnormality. Accordingly, the patient was referred to the orthopedic department for further treatment. However, due to the close proximity of the nodule with the bladder and its special anatomical position, the patient was not treated in the orthopedic department, and was requested to attend regular rechecks. Because the above symptoms became aggravated the patient attended our hospital for further treatment. Bimanual diagnosis: Tenderness in the posterior area of the pubic symphysis was obvious. A pelvic separation test was negative. Reexamination of the pelvic CT and a cystoscopy showed nodular bony processes in the posterior part of the pubic symphysis, which were more obvious than before, with compression of the adjacent anterior wall of the bladder. However, no clear foreign body was observed in the bladder cavity and the mucosa of the protuberant part of the bladder was normal Figs. . The patWe finally decided to perform open pelvic surgery. During the operation, nodular bone tissue from the pubic symphysis was seen to be obviously oppressing the anterior wall of the bladder, surrounded by inflammatory changes that adhered to the serosa layer of the bladder with no clear boundaries Fig. . The locReference to relevant literature published in the last 10 years –8 showedInterestingly, another case of bladder pseudotumor caused by late degeneration of the pubic symphysis has been reported that was very similar to the present case . In thisA previous study reported that about 55.5% of Chinese adult women have at least one lower urinary tract symptom, with an increase in the number of vaginal deliveries and deliveries confirmed to be risk factors for these symptoms . TherefoPathology suggested that urate deposition may have been a possible pathogenic factor in our patient despite her uric acid level being normal. Relevant literature reports that urate deposition is related closely to hyperuricemia and occurs rarely in patients with a normal serum uric acid level. Hyperuricemia often involves distal hand and foot soft tissue and joints and although it is common for uric acid crystals to invade cartilage, this is rare in the pubic symphysis , 12. DueIn conclusion, female lower urinary tract symptoms are often encountered by urologists and the present case provides relevant experience on the diagnosis and treatment of female lower urinary tract symptoms (Additional file Additional file 1. CARE checklist.Additional file 2. Pathological report.


Gastrointestinal stromal tumors (GISTs) are typically solid, sometimes with small cystic areas, but rarely manifest as predominantly cystic neoplasms. In addition, cystic intestinal GISTs with intratumoral abscess formation are rare.We present the case of a 49-year-old male patient with a history of frequent and urgent urination for 2 weeks. Radiologic studies revealed a large cystic mass in the lower abdomen. The patient underwent abdominal laparotomy, which revealed a large cystic mass arising from the distal ileum invading the sigmoid mesocolon and apex vesicae. Partial resection of the ileum along with the tumor and the adjacent bladder was performed. Macroscopic examination revealed that the cystic mass contained a large amount of foul-smelling pus and a tumor-bowel fistula. The final pathology revealed an abdominal stromal tumor. Postoperative recovery was uneventful, and adjuvant imatinib mesylate 400 mg was administered daily. No tumor recurrence or metastasis was observed during the 9-month follow-up period.Fingings of a cystic tumor in the abdomen should raise concern for cystic GISTs. This case report reviews a rare presentation of an ileal giant cystic GIST with atypical intratumoral abscess formation. Complete surgical resection and adjuvant imatinib is still the mainstay treatment for GISTs. Gastrointestinal stromal tumors (GISTs) are the most common mesenchymal tumors of the gastrointestinal tract, accounting for 0.1%–3% of all gastrointestinal tumors . Most GIA 49-year-old man was admitted to our hospital with a history of frequent and urgent urination for 45 the past 2 weeks. He did not have abdominal pain, fever, dysuria, or hematuria. His medical history was unremarkable except for hypertension. Physical examination revealed distention of the lower abdomen. A large, painless, immobile, and hard mass was palpable, having an estimated diameter of 17 cm. Laboratory testing revealed the following: C-reactive protein, 61.50 mg/L ; white blood cell count, 14.94 × 109/L ; neutrophilic granulocytes, 80.8% ; hemoglobin, 101 g/L ; procalcitonin, 0.25 ng/ml ; interleukin-6, 43.49 pg/ml ; prothrombin time, 14.3 s  and D-dimer, 1.60 ug/ml . Routine urine tests, liver function tests, and tumor markers  were all within the normal ranges. Ultrasonography revealed a large cystic mass, measuring approximately 17 cm in diameter, in the lower abdomen in front of the abdominal aorta and iliac vessels. Contrast-enhanced computed tomography (CT) revealed an abdominal cystic tumor with an unevenly thickened wall and mural nodules; air-fluid levels were not observed within the tumor. Enhancement was noted in the cyst wall and mural nodules during the arterial phase . Magnetivia a mucosal fistula  are the second most common type of GISTs in the digestive tract, with 5-, 10-, and 20-year disease-specific survival rates of 84.4, 71.2, and 54.2%, respectively . A retro18F-fluorodeoxyglucose positron emission tomography (18FDG-PET) can be helpful in differentiating malignancy and ruling out metastatic disease as well as monitoring response to molecularly targeted therapy. Moreover, 18FDG-PET is more sensitive for the assessment of early therapy response than morphologic imaging modalities , followed by the epithelial type (5%), and mixed type (9%). Immunohistochemical tests indicated that KIT, CD34, SMA, S-100, and desmin expression rates were 98%, 40%, 34%, 14%, and 0.2%, respectively . MutatioSurgical resection and imatinib are the most effective treatment for primary GISTs. For cGISTs, high attention should be paid intraoperatively to avoid abdominal dissemination due to tumor rupture. Partial resection of the ileum along with the tumor is the standard procedure for ileal cGISTs and lymphadenectomy should not be routinely performed if there is no evidence of lymph node metastasis. It is well accepted patients with high-risk features had prolonged overall survival (OS) with adjuvant imatinib . HoweverWe present the case of a large cGIST located in the ileum with an intratumoral abscess diagnosed through histopathology after surgical excision. For intraperitoneal cystic lesions, especially those suspected to originate from the gut, the possibility of a GIST should be considered before surgery. We have shared our clinical experience to help guide the management of similar cases.


Intra-oral lipomas are benign and relatively rare tumors, although they occur with higher frequencies in other areas, most especially the back, abdomen and shoulders of adultsFemale, 60 year old, with 3 years of accelerated development which caused a rare large size of this type of lesion. Treatment of choice was the surgical removal of this lesion after incisional biopsy of the tumor and diagnostic confirmation of lipoma. The patient is currently under observation after 6 months of follow up, without complications .Figure 1Cases 2 and 3 are related to smaller lipomas, most commonly found in the oral cavity. In both cases, the patients were females, with ages ranging between 30 and 45 years, respectively. In case 2, the lesion was in the inferior lingual region; and in case 3, it was in the jugal mucosa . Both we5.Lipomas are histologically very similar to normal fat tissue1. However, its metabolism is quite different from that of the normal tissue, because its lipids are not available for our normal metabolism5. It more frequently affects persons with more than 40 years of age and bears equal intra-oral gender distributionIt bears variable sizes, from small −10mm masses, to large fat lesions and happen most frequently in the oral mucosa. Tongue, mouth floor, jugal mucosa, vestibule, palate, lips and gums are the most common sites, in descending order5.Although trauma, infection and other factors have been proposed as etiological agents for lipomas, their etiology remains unknown. The treatment of choice is conservative surgical excision and recurrence is rare, as is its malignant transformation


Disseminated peritoneal leiomyomatosis (DPL) is a benign metastasis of leiomyoma mimicking metastasis of malignancy. It usually affects premenopausal women. Malignant transformation is a rare clinical scenario of DPL. However, its etiology is unknown with unusual growth patterns, either of which makes the diagnosis difficult. It was postulated that the pathophysiology of DPL is metaplasia of mesothelial cells under the effect of hormonal stimulation. Hence, we reported the case of a 62-year-old woman with a history of left breast cancer, who presented with DPL and metastasis to the lung with malignant transformation after two years of starting prophylactic tamoxifen therapy. The influence of tamoxifen use on the development of DPL is not fully understood; this is a rare case that highlights a possible association between tamoxifen and the malignancy transformation of DPL. Hence, it may help raise awareness among clinicians dealing with women using tamoxifen or other hormonal therapy, and the risk of DPL development with potential malignant transformation in such patients. Disseminated peritoneal leiomyomatosis (DPL) is a rare benign disease with an estimated prevalence of <1:1000,000. It was first described by Willson and Peale in 1952 . TravassWe report here the case of a 62-year-old postmenopausal female, a known case of diabetes, hypertension, G6PD reduced activity, and breast cancer with left-sided mastectomy four years ago; the postoperative patient was kept on prophylaxis tamoxifen; however, the patient was non-compliant and took tamoxifen irregularly for two years. The patient was referred to gynecology oncology after presenting to the emergency room with progressive abdominal distension and pain for one year. CT scan showed bilateral complex pelvic abdominal mass, most likely ovarian with thickening of peritoneal reflection and nodular deposits, multiple left lung pulmonary nodules. The patient had a history of total abdominal hysterectomy 17 years ago due to fibroid. It was unknown if the ovaries were removed or not. Her surgical records and histopathology report were lost. Hence, an ultrasound-guided biopsy was done for the patient to determine the nature of the masses; a core biopsy was obtained from the abdominal nodules, which showed linear fragments of highly hyaline stromal tissue, no atypia or malignant cell was noted, and the pathologist recommended repeating the sample if suspicion still exists.The patient underwent laparotomy for excision of the pelvic mass. Intraoperatively, a giant free mobile cyst measuring around 28 x 26 cm in size, filled with gelatinous greenish fluid, was found attached to the mesentery; two other masses were also seen, one attached to the omentum measuring 5 x 5 cm, and the other attached to the mesentery measuring around 13 x 12 x 4 cm  with the suspicion of low-grade leiomyosarcoma (LMS). Due to a non-conclusive diagnosis, the specimen was sent to another hospital laboratory for a second opinion. The histopathology report consisted of interlacing bundles of smooth muscle fibers; the cyst that was attached to the mesentery showed a focal increase in mitosis of 4-6/10 HPF and foci of degenerative atypia and no area of convincing necrosis was seen. This is in line with the leiomyoma of the three excised lesions, as the focal area of increased mitosis and degenerative atypia can be seen with leiomyomas. Immunohistochemistry investigation results were positive for SMA, desmin, and estrogen receptors and negative for CD10, CD117, DOG-1, CD34, myogenin, Pan-CK, beta-catenin, and Ki6<10% Figure .Also, the patient underwent left thoracoscopy and wedge resection of the lung including lower lobe superior segmentectomy + partial lingulaectomy of the left lung. The pathology report consisted of circumscribed neoplasm composed of spindle cells arranged in a fascicular pattern with trapped bronchial tubules lined by respiratory epithelium which was positive for TTF1 and CK7. There were mitosis of 5/10 HPF, focal necrosis, focal nuclear polymorphism, and focal tumor giant cells. All resection margins were free from tumors. Immunohistochemically, the lung specimen was positive for smooth muscle actin (SMA), muscle-specific actin (MSA), desmin, and Ki67:10-12% and negative for CD34, DOG1, and C-Kit beta-catenin. The lung lesion specimen consists of LMS. The case was discussed by the National Tumor Board and the patient opted for chemotherapy in another hospital.In this report, we present a rare case of DPL with transformation to malignancy in a postmenopausal woman after 17 years of her hysterectomy. Generally, DPL is usually benign; however, the risk of malignant transformation is 2-5% . It usuaRosatiet al. have reported a case series with a patient affected with DPL and malignant transformation and the diameters of the max lesions were in the range of 10-20 cm . As far DPL's resemblance to malignancy by radiology, grossly and intraoperatively, makes its diagnosis challenging . Even miDPL with malignant transformation is an extremely rare condition, because of which data on the pathogenesis, risk factors, and management of DPL and its malignant transformation remain insufficient .In this report, we highlighted a case of DPL with metastasis and malignant transformation in a postmenopausal woman on tamoxifen therapy. Malignant transformation of DPL is a rare, yet critical condition that is best managed by a multidisciplinary team to achieve an optimum diagnosis and management. Tamoxifen indications were expanded worldwide, especially in patients with breast cancer; nevertheless, industrious surveillance of the adverse side effects is needed. Further research studies will be necessary to address if the association between tamoxifen therapy and DPL exists. As such, this case report will create awareness of this rare event and will help in guiding the providers caring for women using tamoxifen or other hormonal therapy. Furthermore, another hormonal therapy as a selective estrogen receptor modulator without the uterine-proliferative effects, such as raloxifene, may be more suitable for women with a previous history of leiomyoma.