Introduction: Primary hepatic leiomyoma (PHL) is a rare benign hepatic tumor with unclear pathogenesis. It more commonly occurs in immunosuppressed patients, while only 24 cases have been described among immunocompetent individuals. To date, only one successful preoperative diagnosis of PHL has been achieved.Case Presentation: Here we report a case of PHL in a middle-aged woman with no history of immunosuppression. Preoperative diagnosis of PHL was established using ultrasound-guided fine needle trucut biopsy (FNTB). Nevertheless, due to the growing nature of tumor and patient's symptoms, we proceeded with surgical resection, which confirmed the diagnosis of PHL. At 6-month follow up, the patient is in good condition with no evidence of tumor recurrence.Conclusions: PHL is an uncommon tumor that should be considered in the differential diagnosis of rare liver tumors. Image guided FNTB appears to be effective in achieving preoperative diagnosis of PHL. Surgical resection, however, remains both diagnostic and curative in the management of PHL. Leiomyoma is a benign mesenchymal tumor with smooth muscle differentiation that commonly occurs in genitourinary system, particularly uterus, and gastrointestinal tract . PrimaryHere we report a case of PHL in an otherwise healthy, middle-aged female with no history of immunosuppression.A 56-year-old Caucasian female was referred to Clinical Department of Abdominal Surgery, University Medical Center Ljubljana for further investigation and treatment of a growing intrahepatic mass. Initial abdominal ultrasound (US), which had been carried out 6 years previously for evaluation of nephrolithiasis, revealed a hypoechoic lesion in a segment VII of the liver. Further contrast-enhanced computed tomography (CT) scan confirmed a 15 mm hypervascular lesion in segment VII . PeriodiIn her past medical history, she underwent laparoscopic cholecystectomy due to acute calculous cholecystitis. She had no history of chronic illnesses and she took no medications. She reported intermittent pain in the right upper quadrant, which had started few weeks before the admission. Physical examination was unremarkable. Laboratory results including tumor markers alpha-fetoprotein (AFP), carcinoembryonic antigen (CEA), and cancer antigen 19-9 (CA 19-9) were all within normal ranges.Contrast-enhanced US showed a hypoechoic lesion with hyperenhancement in arterial phase that progressed from the periphery to the center and subsequent washout in the portal-venous phase. Further magnetic resonance imaging (MRI) demonstrated the same lesion, which was better defined as a 45 × 49 mm round lesion in segment VII with a hypointense signal on T1-weighted sequences and hyperintense signal on T2-weighted sequences with complete homogenous enhancement in post-contrast arterial phase and hypointense signal in hepatobiliary delayed phase . These fSince imaging studies were inconclusive, US-guided 18G fine needle trucut biopsy (FNTB) was performed. Biopsy specimen, measuring about 50 × 1 mm, revealed a benign mesenchymal tumor composed of bland spindle cells forming short fascicles. The nuclei of tumor cells were elongated and often cigar shaped lacking mitotic activity, while the cytoplasm was moderately abundant and eosinophilic. By immunohistochemistry, tumor cells were positive for smooth muscle actin, desmin, H-caldesmon and calponin, and were negative for CD117, DOG1, STAT6, MUC4, and wide spectrum cytokeratins. Based on the morphological features and supplemented by additional immunohistochemistry, a diagnosis of a benign smooth muscle tumor was suggested, consistent with leiomyoma.Patient's records were presented at a hepatobiliary multidisciplinary team meeting. As tumor had grown progressively to a size of almost 50 mm and the fact that it became symptomatic, surgical removal was recommended. Thus, laparotomy was carried out in an usual manner. Surgical exploration and intraoperative US revealed a 49 × 41 mm solid mass in segment VII of the liver in close proximity to the right hepatic vein. Resection of segment VII  was performed using cavitron ultrasonic surgical aspirator (CUSA) . Furthermore, two simple hepatic cysts in segment II were also removed. Finally, a thorough abdominal and pelvic exploration with gastric and small bowel palpation was performed, all of which were unremarkable. The postoperative period was uneventful and the patient was discharged on the fifth day after surgery.In situ hybridization was negative for Epstein-Barr virus (EBV). In summary, morphological and immunohistochemical features were consistent with hepatic leiomyoma. An extensive clinical work-up failed to detect the presence of smooth muscle neoplasm outside the liver, including gastrointestinal and genitourinary tract. Therefore, a final diagnosis of a PHL was established.Histologic analysis revealed a well-demarcated and unencapsulated tumor within the liver , composeAt 6-month follow up, the patient is in an excellent clinical condition, with no evidence of recurrence. She continues to adhere to surveillance at every 6 months.PHL is an uncommon, benign hepatic tumor that affects both children and adults with slight female predominance . To diagThe pathogenesis of PHL is not well-understood. Although, it was proposed that PHL can originate from smooth muscle cells of hepatic vessels or bile ducts, this has not been proven in later studies , 8. In gPatients with hepatic leiomyoma are often asymptomatic, or they have non-specific symptoms that are similar to those of other benign liver tumors. The most common complaint among symptomatic patients is right upper quadrant pain followed by epigastric pain and diffuse abdominal pain . In two The widespread use of imaging techniques has led to an increased detection of incidental liver tumors. Since differential diagnosis of liver incidentalomas is extremely broad, rare liver tumors remain a diagnostic challenge for most clinicians . On US, via interventional/percutaneous methods have been described. To date, only one successful preoperative diagnosis of PHL has been reported by Sousa et al.  for the publication of any potentially identifiable images or data included in this article.BT has been involved in the management of the patient. BL contributed to the pathological diagnosis. BH wrote the first manuscript. MD, BL, and BT assisted in the preparation of the manuscript. All authors have read and approved the final manuscript.The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.


Struma ovarii is a very rare tumour of the ovary, which is usually benign. It is a solid cystic swelling of the ovary, which is characterised by the presence of histologically detectable thyroid tissue. Confirmatory diagnosis is usually on histopathology after resection of the tumour. It is difficult to identify struma ovarii with radiological investigations alone as it may mimic malignancy. In this case report, we present the case of a 48-year-old woman who had a total abdominal hysterectomy for an incidental finding of right adnexal mass on ultrasound scan following a 10-day history of loose stools and pain in the abdomen. It was ultimately found to be a follicular variant of papillary carcinoma in struma ovarii after a pathological examination. Struma ovarii (SO) is a bizarre tumour of the ovary which is a form of matured ovarian teratoma. It is a variant of dermoid tumours of the ovary, in which the majority of the tissue components consist of thyroid tissue . AlthougThis tumour was first described by Boëttlin in 1889 when he observed the presence of thyroid follicular tissue in the ovaries. It is usually benign but the malignant disease is found in a small proportion of the cases . ReportsA 48-year-old female P2L2) was referred to our hospital for a CT scan after an ultrasound scan elsewhere, which revealed a cystic mass in the right adnexa with septations and solid components. She initially came to the hospital with complaints of loose watery stools for 10 days and lower abdominal pain for seven days. She was on day 10 of her menstrual cycle. She admitted to having regular cycles with normal flow. She was nondiabetic and normotensive. She had low-grade fever however no chills, no lower urinary tract symptoms or previous episodes of loose stools lasting for over three days. On examination, she had pallor and seemed lethargic, but had no icterus or lymphadenopathy. There was mild tachycardia (105/min) while the rest of her observations were stable. On abdominal examination, slight discomfort in the lower abdomen and pelvis region was noted; however, there was no obvious palpable mass. Ultrasound scan done elsewhere showed possible tubo-ovarian mass/abscess which could be infective or neoplastic. Apart from an elevated Ca-125 level (68U/L), all other blood investigations including thyroid profile came back normal. We initially considered a possibility of pelvic TB, but Mantoux test was negative. CT scan revealed lobulated heterogeneously enhancing complex cystic mass measuring approximately 9.2 x 6.8 x 4.3 cm in the right adnexa extending up to the pelvic brim (Figure L2 was reAn ultrasound scan suggested the possibility of a tubo-ovarian mass or an infection, however, infection was unlikely after the CT scan. The absence of areas of calcification or fat density further ruled out the possibility of a mature cystic teratoma, while the presence of soft tissue component in the scan made mucinous cystadenoma a less likely differential. The characteristic feature of a large lobulated multiloculated solid cystic mass showing enhancement made malignant ovarian mass a strong possibility, although the absence of enlarged lymph nodes in the surrounding areas and lack of evidence of metastasis dampened the possibility.The decision was taken for surgical resection of the tumour and confirmation of clinical diagnosis. The resected sample Figure  was sent SO is commonly found in patients of their reproductive age. However, it can be seen in individuals of any age group including children . It compPapillary carcinoma of stuma ovarii is a very rare entity and is usually diagnosed post-operatively after histopathological examination of the resected sample. Here, we present the case of a 48-year-old woman who presented with pain in the abdomen and watery stools for seven days and was diagnosed to have papillary thyroid carcinoma in SO. She underwent a total abdominal hysterectomy after suspicion of malignancy from the elevated serum CA-125 levels and the presence of a lobulated complex cystic mass on CT. There are no definite guidelines for the management of this condition owing to its rarity; hence, therapeutic decisions should be made based on clinical judgement and histopathological reports.


Oro-antral Communication is an unnatural communication between the oral cavity and maxillary sinus and when it fails to close spontaneously, it remains patent and is epithelialized so that oro-antral fistula develops. It is a common occurrence following removal of maxillary premolars and molars because of anatomic proximity of root apices of these teeth and maxillary antrum. Signs and symptoms of oro-antral fistula varies from free escape of fluids, pain, pus leakage, voice alteration, to pan-sinusitis. Several surgical options exist for its management, in particular the buccal fat pad technique, which has proved to be an effective and a reliable technique. We report in this article two-succefull cases of oro-antral fistula managed with buccal fat pad. This epThese complications occur most commonly during extraction of upper molar and premolar teeth (48%). The major reason is the anatomic proximity or projection of the roots within the maxillary sinus. Other causes of OAC/OAF include tuberosity fracture, dentoalveolar/periapical infections of molars, implant dislodgement into maxillary sinus, trauma (7.5%), presence of maxillary cysts or tumors (18.5%), osteoradionecrosis, flap necrosis, dehiscence following implant failure and sometimes as a complication of the Caldwell-Luc procedure . These sThere are many techniques for the closure of oroantral communication including buccal or palatal alveolar flaps and their modifications, various alloplastic materials like gold foil, gold plate, soft polymethylmethacrylate and lyophilized collagen, autogenous bone grafts were also used. The choice of technique depends on the size, the localization, and seniority of the lesion, but also on the surgeon's experience .Among other available methods, the pedicled fat pad is a simple and reliable flap for the treatment of these defects.Here we present two cases report treated successfully with the buccal fat pad (BFP) technique.This case report has been reported in line with the SCARE Criteria .22.1A 57-year-old patient referred by his general dentist to our oral surgery department for air and pus leakage in the oral cavity. The patient had a well-balanced diabetes under treatment. On questioning, the patient reported a history of dental extraction eight months before.The extraoral examination had no particularity. At the intraoral examination, we noted a bad oral hygiene with the absence of 16, 26, 35, 46 and 47. The mucosa around the site of 26 was normal with the presence of fistula detectable with a gutta-percha cone.On the panoramic radiograph, a bone defect was noted making the left sinus communicate with the oral cavity . The CT Considering all these data, the patient was put on antibiotics (amoxicillin/clavulanic acid for 10 days) and a nasal decongestant to treat sinusitis. A surgery with BFP was decided to close the oro-antral fistula.Under local anesthesia, an intrasulcular incision going from 28 to 25 with mesial discharge allowed the lifting of a mucoperisoteal flap and the exposure of the fistula and bone defect A silky cleansing of the site was performed with elimination of the fistulous cord. A saline rinse to cleanse the sinus was also performed.A horizontal incision at the level of the periosteum opposite to 28 was made to gain access to the buccal fat ball, which was dragged into the alveolar defect and then sutured on the bone defect . HermetiAntibiotics were continued for 10 days, along with prednisolone 60 mg/day for 5 days, paracetamol and nasal decongestant.The patient was seen again after 10 days. Healing was uneventful with closure of the OAF. At six months, good healing was noted with a return to normal .Fig. 3In2.2A patient aged 45 presented with lack of healing and air leakage from the extraction site of the first left maxillary molar, one month before extraction.On intraoral examination, an empty tooth socket was noted .Fig. 4InOn the panoramic x-ray, a small bone defect was noticed making the oral cavity communicate with the right maxillary sinus. The CT scan showed a thickening of the right sinus mucosa, poor aeration of the nasal meatus and opacification of the ethmoidal air cells .Fig. 5CTTherapeutic intervention and follow up: The patient was put on antibiotics (amoxicillin/clavulanic acid) for 10 days and surgical management with BFP under local anesthesia was decided in the following days to close the oro-antral communication. After the mucoperiosteal flap elevation, a large bone defect was revealed. The bone defect was closed with buccal fat pad after removing the fistula and rinsing the sinus with saline water. Hermetic sutures were used to suture the BFP over the bone defect  and the Good healing was noted after 10 days and 6 months, without any complications.3as in our case [Oro-antral communications, or perforations that connect the mouth and the sinus, are commonly seen in clinical dental practice, especially after extractions of maxillary posterior teeth. According to the literature, the incidence of OAC has been reported to be as high as 11% ,7. Extraour case .If OAF is suspected, a thorough clinical and radiological examination should be proceeded. OAF acts as a pathway for bacterial and fungal penetration leading to maxillary sinusitis, or pan-sinusitis in 60% of cases ,10. SignRadiographic exams such as panoramic view allow us to see an alveolar defect and Waters’ view to see maxillary sinus infection. The communication between the oral cavity and maxillary sinus can be confirmed with Cone beam computed tomography or CT scan. They also permit to note a thickening of the sinus mucosa or its opacification, the aeration of the nasal meatus or the pathological state of the ethmoidal air cells or other sinuses.Closure of the OAF is very important to prevent any food or saliva accumulation that can cause sinus contamination leading to infection, impaired healing and chronic sinusitis . HoweverMeanwhile, patients with chronic sinus disease that doesn't respond to medications will require surgical intervention such as endoscopic sinus surgery or the Caldwell–Luc procedure .Closure of OAF should be based upon certain factors that not only influence the ultimate outcome of surgical closure but also post closure rehabilitation . Some faAmongst the known techniques for closure of OAF are buccal advancement flaps, palatal advancement flaps, rotational advancement flaps, hinged flaps, island flaps, and buccal fat pad . In our It has been a popular method for the closure of oro-antral communications, as a single layer (Stajeic), with free skin grafts (Egyedi) or even covered by lyophilized porcine dermis (Fujimura). Heister ,15 firstThe BFP consists of a main body and four extensions: buccal, pterygoid, superficial and deep temporal. The body is centrally positioned. The buccal extension lies superficially within the cheek, and the pterygoid and temporal extension are more deeply situated .The surgical technique consists of a circular incision with 3mm margins, made around the OAF, epithelial tract and inflammatory tissue within the opening are completely removed. Two divergent cuts are then made from each end of the circular incision extending into the vestibule. The trapezoidal buccal mucoperiosteal flap is then reflected from the alveolar process and the lateral wall of the maxilla. The BFP is exposed through 1cm long vertical or horizontal incision in the reflected periosteum posterior to the zygomatic buttress. The BFP is dissected and gently advanced into the bony defect and sutured to the palatal mucosa without tension. Finally, the mucoperiosteal flap is replaced in its original position, and sutures are placed between BFP and the buccal flap so that a part of BFP is exposed into oral cavity. Epithelization of the exposed fat tissue occurs between 2 and 4 weeks postoperatively. The superficial layer of fat tissue becomes replaced by granulation tissue and is finally covered with stratified squamous epithelium migrating from the margin of the gingival . The BFPRegular post-operative follow-up of the patient is recommended for up to one year to avoid surgical failure and recurrence. Perfect healing was noted in our cases after more than six months of follow-up.This technique presents many advantages, which are high success rate, wide applicability, simple procedure, done under local anesthesia, no additional removal of bone or tooth, low rate of complications, decrease risk of infections, can easily be trimmed to appropriate shape and no loss of sulcus depth. Its disadvantages are single use, the possibility of postoperative trismus, limited use for small and mid-sized defects and no rigid support .4Oral-antral fistula are frequent complications in dentistry. Their diagnoses require careful clinical and radiological examination. An early closure of post-chirurgical AOC is the best way to prevent AOF and sinus complications. Buccal fat pad represents a reliable treatment option that is easy and predictable for the closure of AOF.No source has funded this manuscript.All the authors have read and complied with the policy of the journal on ethical consent.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.Dr MAINASSARA CHEKARAOU Samir designed the concept, analyzed and interpreted the findings, wrote and reviewed the final paper under the supervision of Prof Laila Benjelloun and Prof El Harti Karima.Mainassara Chékaraou Samir.Not appliableThe authors declare that they have no conflict of interest.


A 61-year-old, otherwise healthy man presented to our hospital complaining of intermittent colicky pain of left-sided abdomen, bloating, and constipation for the prior month. He had no abdominal surgeries or significant problems in his past medical history. Physical examination revealed mild anemia and abdominal tenderness. The patient’s hemoglobin level was 11.8 g/dL and his carcinoembryonic antigen level (CEA), erythrocyte sedimentation rate (ESR), and C-reactive protein (CRP) were normal.Contrast-enhanced abdominal computed tomography (CT) revealed the presence of a concentric mass in the descending colon with a density of approximately -100 Hounsfield units . A colonWhat is your diagnosis?Answer: Descending colon lipoma. Pathological examination of the surgical specimen showed mature adipocytes in the submucosa with mucosal ulceration with no signs of malignancy.Lipomas of the gastrointestinal tract are rare non-epithelial tumors and most often discovered incidentally during colonoscopy, imaging, surgery, or autopsy. Colonic lipomas are more prevalent in women and have a peak incidence in patients between the 4th and 7th decade of life; their incidence ranges from 0.2% to 4.4%. Common sites of colonic lipomas are the ascending colon (45%), sigmoid colon (30%), descending colon 15%), and transverse colon (9%), respectively  is a valuable non-invasive imaging method for the diagnosis of colonic lipomas. The characteristic sign is an intraluminal mass with regular, ovoid, and sharp borders at a homogeneous fat density. In MRI, the signal intensity of adipose tissue and suppression of signal in fat-suppressed sequences have been seen; therefore, MRI may be especially beneficial in the detection of lipomas .Colonoscopy is suggested when a diagnosis of colonic cancer is suspected. Three endoscopic signs can aid: (a) the “cushion” sign, which occurs when forceps press into the mass, resulting in pillowing; (b) the “tenting” sign, which occurs when mucosa is grabbed and pulled away, resulting in a tent-like appearance; (c) the “naked fat” sign, which occurs when fat is extruded after biopsy (1). In some cases, endoscopy may reveal ulcerative lesions of the polypoid mass leading to a presumption of malignancy. Endoscopic ultrasonography can help demonstrate the presence of a hyperechoic mass. Evaluation of the tumor’s base is the key of indication for endoscopic resection. There are several techniques for endoscopic removal, including endo-loop, nylon loop, endo-clip resection, and sectioning of the overlying mucosa via segmental cuts .Surgery is the standard therapeutic option for lipomas with a diameter greater than 2 cm. It may also be warranted in cases of unclear preoperative diagnosis, symptomatic patient, involvement of the muscular or serosal layer, and when the lesion cannot be radically resected endoscopically . LaparosThe authors declare that they have no conflict of interest.


Pelvic inflammatory disease (PID) and tubo-ovarian abscess (TOA) are serious diagnoses to consider in reproductive-age women presenting with abdominal or pelvic pain. Management can be medical or surgical depending on severity. This case report outlines the unique presentation of TOA in a 15-year-old female presenting with acute abdominal pain with a recent past surgical history of appendectomy. A discussion of the approach to similar presentations and the importance of maintaining a broad differential diagnosis follows. E. coli, B. fragilis, Prevotella, and Peptostreptococcus. The differential diagnosis of a TOA includes ovarian torsion, ectopic pregnancy, appendicitis, gastroenteritis, constipation, and urinary tract infection (UTI).A tubo-ovarian abscess (TOA) is a feared complication of pelvic inflammatory disease (PID) involving abscess formation in the uterus, fallopian tube, and ovaries. Infection can develop due to an ascending vaginal infection or from a gastrointestinal infection as not all cases of TOA are associated with PID. Between 15-35% of women treated for proven PID will also be diagnosed with TOA . TOAs teManagement of TOA may be medical, surgical, or both and should be aggressive as the risk of delayed treatment can predispose to rupture or sepsis. Medical management usually involves intravenous (IV) antibiotics with an empiric regimen selected on the basis of most likely organisms. Patients are usually placed on two or three agents to allow for adequate coverage. Unfortunately, intravenous antibiotic therapy can have limited efficacy with large abscesses due to poor penetration into the target tissue. Progression to surgical treatment is necessary after poor response to antibiotics or in the setting of hemodynamic instability at presentation. Minimally invasive approaches are emphasized depending on pre-operative imaging findings. Open techniques can be considered for complicated pathology or large abscesses.A 15-year-old nulliparous female with noncontributory past medical history presented to the emergency department (ED) at our institution with acute onset abdominal pain that worsened over two days. She rated her pain as 5/10 and denied nausea, vomiting, diarrhea, fever, and chills. A recent appendectomy was performed five months ago which was complicated by postoperative peritonitis. At that time, she developed a transaminitis likely secondary to an inflammatory process resulting from her acute suppurative appendicitis. Her maximum alanine aminotransferase (ALT) was 149 and her maximum aspartate aminotransferase (AST) was 184 with an elevated C-reactive protein (CRP) of 4.2. She required IV metronidazole and piperacillin/tazobactam followed by an outpatient course of cefdinir and metronidazole for ten days. Her total duration of antibiotic therapy post-surgery was 14 days. She recovered after the appendectomy and did not have any further post-operative issues until her current presentation to the ED for abdominal pain. Her only previous surgery was the appendectomy five months prior. She does not take any medications and has no known drug allergies. She denies smoking, alcohol use, and drug use. She also denies sexual activity. Her family medical history is noncontributory.Her vitals in the ED were stable with a temperature of 99.6 F. She was alert and oriented without acute distress. Her cardiac and lung exams were within normal limits. Upon abdominal exam, she was tender to palpation with positive guarding and rebound. She had active bowel sounds and no costovertebral angle tenderness. There was no edema of the extremities or calf tenderness. Her distal peripheral pulses were 2+ bilaterally.Her complete blood count (CBC) and complete metabolic panel (CMP) drawn in the ED were within normal limits. Urinalysis revealed 1+ leukocytes, 1+ protein, and all other findings within normal limits. Ultrasound in the ED showed a complex right annexation lesion adjacent to the right ovary measuring 7.1 cm x 4.3 cm x 4.3 cm with heterogenous echogenicity and a central anechoic fluid collection  with a pre-operative diagnosis of a hemorrhagic ovarian cyst. A diagnostic laparoscopy was performed with drainage of the right pyosalpinx and subsequent removal of the stump appendix by general surgery. The stump appendix was evaluated by pathology and found to be a portion of benign appendix without evidence of neoplasia or an active inflammatory process. The pyosalpinx was comprised of necrotic material with acute inflammatory debris and no evidence of neoplasia. Other findings observed during the surgery included purulent fluid in the cul-de-sac, a normal right ovary, a normal left fallopian tube, and a normal left ovary. The post-operative diagnosis was amended to right pyosalpinx and stump appendix.Post-operatively the patient did well. Her pain was controlled, and her Jackson-Pratt drain had minimal serosanguinous output. Antibiotics were continued, including doxycycline, metronidazole, and cefoxitin. Her diet was advanced to regular. She voided after the foley catheter was removed and passed flatus. She was discharged to home and followed up in the outpatient setting.At the first follow-up appointment, the patient was again asked about her sexual history. She denied previous sexual activity. A cervical culture procured at this time was negative for gonorrhea and chlamydia.Acute abdominal pain in the female pediatric population requires consideration of a broad range of differential diagnoses involving many organs including but not limited to ovaries, uterus, stomach, small and large bowel, appendix, bladder, ureters, kidneys, pancreas, liver, gallbladder, spleen, lungs, and neurologic or psychiatric causes. TOA is an urgent issue that cannot be missed and requires prompt initiation of intravenous antibiotic therapy due to both short-term complications such as peritoneal spread and systemic inflammatory response progressing to sepsis and long-term complications like chronic pelvic pain and future infertility . EmpiricOur patient denied any history of sexual activity when interviewed in the inpatient and outpatient settings. However, it is reasonable to consider that an adolescent may not be forthcoming about sexual history especially in the presence of a parent for fear of punitive familial repercussions. Therefore, it is possible that the source of infection for this TOA was a sexually transmitted infection (STI) leading to PID. Furthermore, a cervical culture was not taken from the patient until her outpatient follow-up appointment approximately one week after discharge from the hospital. It is possible the antibiotics she was prescribed eliminated any evidence of STI that may have previously been present. The 2015 Centers for Disease Control and Prevention guidelines recommend parenteral inpatient treatment with a second- or third-generation cephalosporin, doxycycline, and metronidazole for severe pelvic inflammatory disease requiring inpatient admission .Cases of TOA from hematogenous spread of gastrointestinal pathology and subsequent seeding of ovarian tissues have been described in the literature but are exceedingly rare . Given tThe prognosis of TOA is variable ranging from no complications to recurring PID to development of pelvic pain or infertility . The risTOA is a potentially dangerous complication of PID with many other conditions on the differential in a young female patient. This case of a 15-year-old female with abdominal pain and a recent surgical history illustrates key points in the diagnosis and management of TOA. It is generally advised to maintain a broad differential diagnosis especially in the setting of recent surgery or unclear social history. As TOA can be a potentially fatal condition, it is important to maintain a high index of suspicion in key demographic groups especially given the myriad ways in which the condition can emerge, including ascending vaginal flora, STI, and gastrointestinal seeding.


Synovial chondromatosis is a non-cancerous tumor characterized by the formation of multiple nodules of cartilage due to metaplastic development of the synovial membrane. Etiology can be a primary lesion, of which pathogenesis remains unknown, or low-grade trauma or internal disorders. This pathology can long remain undiagnosed and leads to therapeutic wandering, especially since clinical manifestations are non-specific. Symptoms may mimic temporomandibular disorders and dental orthopantomogram does not always show intra-articular calcified bodies. Cone beam computed tomography (CBCT) and magnetic resonance imaging (MRI) are tests of choice for the diagnosis of this pathology. This case report describes the clinical manifestations, diagnosis and management of a case of synovial chondromatosis involving the temporomandibular joint, in a 21-year-old woman who was initially treated for two years for a common temporo-mandibular disorder. The evidence gathered during the medical interview and clinical examination led us to suspect synovial chondromatosis of the temporomandibular joint. Prescription of a CBCT and MRI confirmed the diagnosis of her temporomandibular joint disorder and led to a successful arthroplasty. Synovial chondromatosis (SC) is described as a non-cancerous tumor arising in the lining of a joint with formation of benign nodular cartilaginous proliferations from the synovial tissue. These nodules may loosen inside the articular space and may cause chronic pain and swelling in the preauricular region when the temporomandibular joint is involved . SC mainSynovial chondromatosis was first classified in three classes by Milgram in 1977.Early stage (Class I) is characterized by synovial symptoms without any loose bodies visible on X-Rays.Intermediate stage (Class II) reunites both clinical symptoms and images of calcified loose bodies within the synovial membrane.Final stage (Class III) is characterized by calcified loose bodies without any synovial involvement .This classification was completed by Gerard et al. in 1993, with criteria based on the calcification degree of the loose bodies . Then, iDifferential diagnosis between temporomandibular disorders and synovial chondromatosis may be difficult and challenging, especially when the patient is young. In this context, we report the case of a synovial chondromatosis of the TMJ, in a 21-year-old woman, which caused intense orofacial pain. Practical recommendations, in terms of clinical and radiological examination, to avoid diagnostic and therapeutic wanderings, are also given.A 21-year-old woman came to the general dental consult of Toulouse’s Teaching Hospital (France) for localized pain involving her left temporomandibular joint. She reported intense, persistent pain, increasing during chewing and/or muscle fatigue, associated with joint noises described as “squeaking”. She explained being followed for the last 2 years for osteoarthritis of the left temporomandibular joint associated with myalgia. For this, the patient had benefited from maxillofacial physiotherapy sessions and the use of an occlusal splint associated with a behavioral approach in the context of her chronic pain. Despite the treatment, she showed no improvement and consulted for further advice in our dental hospital. The semi-directive interview revealed a history of whiplash that had occurred several years earlier, without head trauma. Clinical examination, based on Diagnostic Criteria for Temporomandibular Disorders (DC-TMD) , revealeThe dental orthopantomogram was not very contributive regarding lesions of the left condyle. No joint pinching, enlargement, or lysis of the condylar head was found ruling out the unlikely diagnosis of osteoarthritis . However2) were removed from the synovial lining. Anatomopathological analysis carried on these calcified bodies confirmed the diagnosis of synovial chondromatosis. These nodules are composed of a locally mixoid cartilaginous framework and punctuated with calcium deposits. Some bodies were developed under the synovial intimal layer, thus confirming the diagnosis of class II of Milgram.About thirty chrondroid fragments varying in size from 1 to 7 mm  were prescribed in order to recover correct oral opening and ensure the normalization of occlusal contacts.Primary lesions, of which pathogenesis remains unknown. Cartilaginous metaplastic proliferation of fibroblasts is a hypothesis and could cause primary lesions, without any well-identified genetic or risks factors .Secondary Synovial chondromatosis following, infection, embryological disturbances or trauma such as repetitive low-grade trauma, internal derangement, parafunctions, or even degeneration ,12. SynoEtiologies of synovial chondromatosis are still unknown and are discussed in the few publications on the subject. Suspected etiologies can be divided into two categories:The study of the recent scientific literature gives no precise prevalence of synovial chondromatosis of the temporomandibular joint. In general, SC affects men more frequently than women except for the temporomandibular joint which is more frequently affected in women with a 4:1 sex ratio . DiagnosNevertheless, as soon as Cone Beam-Computed Tomography (CBCT), CT-scan or Magnetic Resonance Imaging (MRI) are performed, loose calcified and/or non-calcified bodies can easily be identified, with or without extra-articular extensions ,19. Lee Diagnostic wise, TMJ arthroscopy is a real improvement and can also be used for treatment . Brabyn Open surgery is indicated in case of invasive SC into adjacent tissues  or if free bodies are present in the lower compartment ,24,25,26Although diagnosis on the basis of clinical elements can be difficult and confusing with TMJ disorders, the therapeutic approach is, at present, widely proven. Open arthrotomy with removal of loose bodies is the gold standard in terms of treatment for this disease. Synovectomy, with or without condylotomy or condylectomy can be associated to this procedure if necessary . Oral–faFinally, the recurrence rate seems to be low. Cases of recurrence have been described and have required larger resections that can go as far as condylectomy and require reconstruction ,27.Concerning the clinical case presented, the one-year clinical follow-up of the patient showed clear improvement of the oral opening and the absence of painful symptoms. In addition, the presence of a Milgram stage 2 requires radiological monitoring for possible recurrence due to the possible persistence of metaplastic foci.Synovial chondromatosis is a relatively rare pathology when it is located at the temporomandibular joints. Clinical signs being non-specific, it can be mistaken for more common temporomandibular disorders whose prevalence are greater. The appearance of pain in the temporomandibular joint with an oral opening limitation in young adults, and more particularly in young women, must alert the clinician, especially if whiplash, even several years back, is mentioned during the anamnesis. Cone beam-computed tomography or magnetic resonance imaging will make it possible to make an early and accurate diagnosis, thus avoiding therapeutic wandering and implementing appropriate management with maxillofacial surgery centers.


Cotyledonoid leiomyoma is an unusual uterine myoma due to some ultrasound features that mimic a malignant lesion facilitating the choice of radical surgery. This study aims to summarize the ultrasound and the magnetic resonance imaging aspects of this atypical lesion, and also discuss surgical treatment and pathological exam. It included all English case reports or case series until August 2021 found through PubMed, Google Scholar, and Scopus. A total of 94 cotyledonoid leiomyomas were reported, with a median tumor size of 12 cm. The typical ultrasound image is characterized by a large solid heterogeneous mass, with high vascularity, no shadowing, and indistinct margins within the myometrium. Magnetic resonance imaging shows the presence of merging isointense nodules to the myometrium in T1-weighted images, hyperintense in T2-weighted images, and contrast agent enhancement. Surgical treatment consists of hysterectomy  or myomectomy , without evidence of recurrence if complete. The placenta-like appearance observed during surgery supports this rare fibroid hypothesis. The intraoperative frozen section can be considered. Microscopically, no atypical cells, signs of mitotic activity or cell necrosis are found. To conclude, some preoperative and intraoperative aspects of this lesion are distinctive and may lead surgeons to opt for conservative surgery. Cotyledonoid leiomyoma is a rare variant of uterine myoma that macroscopically resembles placental cotyledons . Its larHistology is the most accurate method to reach a definitive diagnosis for cotyledonoid leiomyoma. So far, no example of malignant behavior or recurrence have been reported in the typical form of cotyledonoid leiomyoma. However, histological variants will have to be placed in the proper context for these patients’ optimal management. For instance, identifying variants with a more aggressive biological behavior and a potential vascular invasion tendency would be significant to guide the follow-up. Awareness of cotyledonoid leiomyoma variants is necessary for an accurate diagnosis and facilitates appropriate management decisions at the time of surgery. Interestingly, increasing evidence suggests that ultrasound and magnetic resonance imaging features, alongside the bulky mass placenta-like appearance during surgery, can substantially improve diagnosis quality. In particular, imaging evaluation can be an asset in managing patients who are appropriate candidates for fertility-sparing surgery and desire this approach. Indeed, imaging analysis would help evaluate the intrauterine tumor resection by myomectomy and the extrauterine tumor by excision. Based on a recently published case report series , whilst An English literature search was conducted from inception until August 2021 in the following databases: PubMed (Medline), Google Scholar, and Scopus. “Cotyledonoid leiomyoma”, “cotyledonoid dissecting leiomyoma”, “grape-like myoma”, and “Sternberg tumor” were the search terms, and full-text articles were obtained to identify potentially eligible studies. Case studies and case series were included, while literature reviews and cases reported several times by the same authors in subsequent publications were excluded to avoid the same case being considered twice. Clinical and laboratory data were then studied, and the most significant imaging aspects were revised. Pelvic ultrasound and magnetic resonance imaging, surgical treatment, and pathological diagnosis were briefly discussed, based on all the available data. Limited data were achieved in most case reports’ ultrasound and resonance features; therefore, we reported all the cumulative characteristics described. A total of 94 cases with a histologically proven diagnosis of cotyledonoid leiomyoma  were collected, including our 13 cases recorded at the IRCCS Burlo Garofolo of Trieste and the Aviano Regional Cancer Centre [The median age was 44 (range 21–73 years), covering the different ages of the women. Presenting symptoms were the same as for typical fibroids, such as the presence of abdominopelvic masses , abnormal uterine bleeding , abdominopelvic pain , or abdominal swelling , but cotyledonoid leiomyoma were also found incidentally . Serum CA-125 was reported within normal limits or slightly increased [Many cases described in the literature report lesions located on the lateral wall of the uterus, although they can develop as a myoma in all uterine parts, even in the cervix and in the ovary. Because they often extend into the broad ligament and thus into the pelvic cavity, the suspicion of malignancy is even greater, especially in cases where the leiomyoma has a dissecting character. Although considered benign tumors, the macroscopic appearance of these lesions associated with characteristic ultrasound features may induce malignancy suspicion.The gynecologist can come across a heterogeneous irregular large mass with a high vascularization ,42, no sIf required in the suspicion of infiltrative disease, pelvic magnetic resonance imaging can reveal a lobulated mass that shows isointensity to the myometrium in T1-weighted images, mostly hyperintensity in T2-weighted images, and contrast agent enhancement ,26,34,41Our data show that the majority of patients underwent a non-conservative treatment with a hysterectomy (80%), while simple tumor resection or myomectomy was carried out in only 19 patients (20%). The pre-surgical identification of a cleavage plan can guide the choice of the treatment. The cotyledonoid leiomyoma appears to have a capsule that creates the cleavage plane with the myometrium and thus effectively makes myomectomy possible. On the other hand, the capsule is present by ultrasound also in the cotyledonoid dissecting leiomyoma, but it is interrupted where it dissects the myometrium. Although myomectomy is easier in non-dissecting forms, even in the dissecting types it is feasible, regardless of the large size of the neoformation ,45, as sHormone therapy with UPA  or GnRH analogues has been described before myomectomy, resulting in improved symptoms and in reduction in mass volume ,42, but Given the good outcome reported in cases treated with myomectomy, treatment options should be oriented to the simple resection of the tumor, especially in patients who are candidates for fertility-sparing surgery, while hysterectomy should only be considered if needed. Sometimes choosing minimally invasive surgery can be extremely difficult because the intraoperative macroscopic aspect and the preoperative ultrasound features may raise the suspicion of malignancy, inducing the surgeon to perform radical surgery.If recognized, some preoperative and intraoperative elements may help to correctly identify the type of the lesion . In situThe differential diagnosis must consider benign pathologies , and malignant ones. The comparison and differentiation of cotyledonoid leiomyoma from uterine sarcoma is the actual difficulty. Uterine sarcoma may have a rapid growth and ultrasound images can show an intense vascularization with irregular distribution of vessels and several anechoic areas due to cell necrosis . The magThe pathological study of the cotyledonoid leiomyoma reveals only benign aspects . DespiteImmunohistochemistry can confirm the benign leiomyomatosis nature of the lesion . The tumCotyledonoid leiomyoma can have a variety of histological variants, the natures of which are not always predictable. In addition to the dissecting form, the following variants have been identified from the cases described so far: (1) 4 with intravenous leiomyomatosis , (2) 6 wAmong these forms, Niziurski et al.  describeSo far, only Kashima et al. in 2019  have repNon-neoplastic cystic lesions within the cotyledonoid dissecting leiomyoma were also reported (7 cases), including in cases associated with adenomyosis ,46, endoKnowledge of this unusual form of fibroid and its dissecting variant is crucial in managing the lesion, mainly as it avoids unnecessary extensive surgery. In our opinion, the most significant elements in the diagnosis of cotyledonoid leiomyoma are the typical ultrasound, magnetic resonance imaging features, and the placenta-like appearance of the bulky mass during surgery.Further studies are needed to clearly define all variants of cotyledonoid leiomyoma, including their immunohistochemical expression. These insights will undoubtedly make it possible to comprehensively establish the possible vascular invasion tendency, allowing a more specific treatment for those variants.


Metastatic deposits are the most common cause of pleural masses, solitary or multiple. Primary pleural neoplasms are rare entities that are occasionally encountered. Of these, the tumors of neurogenic origin are exceedingly rare with only a few cases reported in the literature. We describe a case of an isolated neurofibroma involving the pleura in a 23-year-old male patient, who underwent CT and MRI scans of the thorax as part of the initial investigations. The final diagnosis was clinched by a CT-guided biopsy of the lesion.  While pleura remains an important site for metastatic deposits for intrathoracic malignancies, primary pleural neoplasms are rarely encountered . Of thesNeurofibromas are benign nerve sheath tumors that can involve peripheral nerves at any level. They can attain large sizes and pose a diagnostic challenge, especially when present in an unusual location. A search through the literature reveals three case reports of primary pleural neurofibroma without any association with neurofibromatosis.A 23-year-old male patient presented to the outpatient department with complaints of left-sided chest pain and difficulty in breathing, which was gradually increasing for one year. There was no significant past medical history. The patient was a non-smoker. System-specific examination revealed reduced air entry on the left side. The rest of the physical examination was unremarkable. For further evaluation, contrast-enhanced chest CT was done, which revealed two closely abutting mildly enhancing soft tissue attenuation mass lesions in the posterior mediastinum on the left side Figure .The lesions were located between the visceral and parietal pleural layers with associated pleural effusion Figure . The larFor confirming the same, the patient underwent MRI. The lesions were hypointense on T1 weighted images and hyperintense on T2 weighted images Figure . No commA CT-guided biopsy was done. Histopathology revealed fascicles of spindle cells and fibrillar eosinophilic cytoplasm. Immunohistochemistry markers study suggested features of neurofibroma. The patient underwent surgical excision of the mass. Intraoperatively, the masses were found to be pleural-based with smooth, encapsulated with well-circumscribed margins Figure . It was The major differentials for a benign pleural mass include solitary fibrous tumor, benign mesothelioma, mesothelial cysts, lipoma, and calcifying fibrous pseudotumor, with solitary fibrous tumors being the commonest.Neurofibromas are the most common benign peripheral nerve sheath tumors. They can have localized, diffuse, or plexiform growth patterns. The localized form can occur sporadically. However, diffuse and plexiform forms are associated with neurofibromatosis type I. While mediastinal involvement is common in both adults and children , pulmonaThe pleura is a serous layer that covers the thoracic structures and is divided into visceral and parietal pleurae. The visceral pleura is innervated by the vagus and sympathetic trunks while the parietal pleura receives its nerve supply from somatic, sympathetic, and parasympathetic fibers, via the intercostals nerves. The diaphragmatic pleura is supplied by the branches of phrenic nerves. Nerve sheath tumors can arise from these nerves with direct pleural involvement. Langman et al.  reportedSchwannoma is another benign peripheral nerve sheath origin tumor with a spindle cell morphology on histopathology. Outside the central nervous system, the flexor surfaces of the extremities or nerve roots are common locations of occurrence. Few cases of pleural schwannomas have been reported -9. Presenting symptoms largely depend on the size and location of these tumors and are non-specific . Those associated with neurofibromatosis will show other systemic manifestations of the syndrome. Pleural effusion mainly occurs due to mass effect and resultant lymphatic obstruction. Cross-sectional imaging plays a crucial role in the diagnosis and management of pleural masses. Careful assessment of the nature of the lesion, origin, and ancillary imaging findings are useful in narrowing the differentials. Localized neurofibromas are best treated with complete surgical resection. To prevent reoccurrence, tumors should be completely excised. Primary pleural neoplasms of neurogenic origin are exceedingly rare. While histopathology is the mainstay for diagnosis, detailed imaging has a significant impact on management. This case report emphasizes the need to be aware of the possibility of such tumors in an uncommon location and of adequate imaging studies before the operative management. Imaging not only delineates the origin but also reveals the relation of the lesion with adjacent structures. Tumors in the paraspinal region of the posterior mediastinum can be easily be confused with a pleural tumor, particularly in a large lesion, where the origin is often obscured. Hence, MRI, in adjunct with CT, is an important modality to assess the intra-spinal extension.


Myopericytoma is a rare, benign growth characterized by painless lesions with a predilection for the extremities, although they may be found in or on any part of the body. These tumors typically present as a rounded or dome-like non-exophytic lesion and exhibit a benign disease course. Treatment is generally reserved for cosmetic or functional purposes. We present a case of an atypical presentation of an exophytic digital myopericytoma in a 45-year-old female treated with local punch excision. Myopericytoma is a rare, benign neoplastic growth, first described in 1998 as a tumor of pericytic/myoid origin . The cauA 45-year-old female presented with an exophytic growth on her left index finger that was present for several years. She denied any pruritis, pain, or bleeding. The patient tried several over-the-counter wart treatments for the growth with no response. She mentioned that it interfered with her ability to type on a computer keyboard efficiently. On physical examination, a 5 mm exophytic pink papule with a hyperkeratotic rim Figure , B was nA punch biopsy of the lesion was performed for histopathological analysis. Immunohistochemical analysis showed that the tumor was focally positive with SMA Figure  but was The clinical presentation described in this case brings to mind several diagnoses, including an acral fibrokeratoma, myofibroma, myopericytoma, myrmecia wart, or less likely a glomus tumor. Often, these entities must be distinguished based on their histopathologic and immunohistochemical features. A myopericytoma is a rare neoplastic growth that can be found anywhere on the body, including the kidneys or brain, but its predilection is more common to the skin of the extremities. Typically, these growths are painless, although painful tumors have been rarely reported . MyoperiThe treatment for a myopericytoma is either observation or complete excision . AlthougOur patient’s hyperkeratotic and exophytic presentation, as opposed to a more rounded, dome-like, subcutaneous, or non-exophytic lesion seen in the limited number of cases published on cutaneous myopericytoma, made this lesion clinically challenging to diagnose. However, histopathology analysis provided the means to make the diagnosis. This case serves as a reminder to clinicians of the varying clinical characteristics that can be seen in these lesions, in addition to the typical histopathology and immunohistochemical staining patterns. Knowledge of this lesion can lead to more efficient diagnosis and treatment plans.


Ectopic teeth in the nasal cavity are a rare phenomenon. They are often associated with a variety of symptoms and future complications, ranging from nasal crusting and obstruction to chronic infections. In most reported cases, their removal is recommended. Here, we report a case of an ectopic intranasal tooth in a symptomatic adult. The tooth was removed endoscopically with good results. The presence of an intranasal tooth is uncommon. Ectopic intranasal teeth may be permanent, deciduous, or supernumerary . The estA 39-year-old female, with a history of chronic rhinitis, presented in June 2020 after several years of left-sided green nasal crusting and discharge. She denied sinus pain, epistaxis, or difficulty with nasal breathing. The patient ultimately came for evaluation because a family member commented on a bad smell from her nose.Of historical significance, the patient recalled breaking her nose and knocking out her front teeth in a roller-skating accident as a young child. The patient could not recall whether this dental trauma affected permanent teeth or deciduous teeth, and she did not remember which front teeth were involved. No history of maxillofacial surgical intervention or further trauma was reported, and past medical history was otherwise noncontributory.Nasal endoscopy revealed a pearly white, hard mass extending from the floor of the left nasal cavity. Gentle palpation of the mass demonstrated mobility. The tip was seen to abut against the left inferior turbinate Figure . AdditioComputed tomography (CT) imaging of the sinuses confirmed the presence of an ectopic tooth, with a poorly-developed crown in the incisive canal and the root extending into the nasal cavity Figure . The tooThe patient underwent endoscopic removal of the intranasal tooth, septoplasty, and inferior turbinate reduction under general anesthesia. The tooth was gently loosened from its soft tissue attachment and removed with a Blakesley forceps Figure . CurettaThe removed tooth was approximately 1.6 cm in length and incompletely developed Figure . All surPostoperatively, the nasal mucosa healed well, with no further crusting, and resolution of symptoms. The patient had no postoperative dental paresthesia.The cause of ectopic tooth eruption and growth is not well understood. Reports of some ectopic teeth have been etiologically linked to displacement by trauma or cysts, while others are associated with congenital conditions, including cleft palate and Gardner syndrome ,4. AlthoRegardless of the etiology and despite its rare prevalence, identifying an intranasal ectopic tooth is necessary in order to resolve symptoms and minimize potential complications. Although sometimes asymptomatic, an intranasal tooth left alone could potentially serve as a nidus for infection, mineralization, and debris , given iSurgical removal, guided by either endoscope or headlights and nasal speculum, is a safe and effective intervention ,3,4,6,8.An ectopic intranasal tooth is a rare phenomenon. While its etiology is not well understood, it is often associated with trauma or congenital conditions. Regardless of the cause, the presence of an intranasal tooth may result in a variety of symptoms and potential complications. An endoscopic removal is a viable option that may eliminate further risks, with minimal harm to the patient.


Parathyroid adenoma is a tumor composed of increased parenchymal tissue, often built-up by chief cells, transitional cells or oncocytic cells arranged in acinar or solid formations. Occasionally, rare histological patterns are reported, including cystic or trabecular arrangements. We present a 47 year-old male patient with primary hyperparathyroidism who underwent focused parathyroidectomy for a right inferior adenoma. Surgery was uneventful, but histologically, normal parathyroid tissue adjacent to a tumorous structure displaying a cystic growth pattern was detected. The cells lining the cyst walls appeared cylindrical and pseudo-stratified, vaguely reminiscent of a respiratory type of epithelium usually associated to branchial cleft cysts or thyroglossal cyst remnants, albeit with a tumorous appearance. The respiratory-like epithelium stained positive for parathyroid markers PTH and GATA3, thereby confirming them as parathyroid-derived. The patient was cured from surgery as he displayed normal calcium and PTH levels postoperatively, and is currently alive and well without signs of relapse 4 years after surgery. This is to our knowledge the first report of a parathyroid tumor displaying a respiratory-like epithelium. Experimentally, canine parathyroid glands can develop ciliated respiratory epithelium in response to inhalation of ozone. Our patient is a construction worker with a hypothetically increased risk of continuous ozone exposure. Although this association remains purely speculative, future investigations of this tumor phenotype could perhaps yield novel insights regarding the frequency of this histological variant, potential clinical associations, and clues regarding influencing factors. Parathyroid adenomas are benign, parenchymal tumors that often secrete parathyroid hormone (PTH), causing hypercalcemia . The mosThe parathyroid chief cells, the main cell type of most parathyroid adenomas, are usually polygonal with a neutral to slight eosinophilic cytoplasm, and the nuclei are often round with little atypia . In normThe patient was a 47 year-old male of Swedish ethnicity without previous medical history. He had no family history suggestive of parathyroid disorders. He worked as a construction worker, did not smoke and only reported moderate use of alcohol. In 2017, he developed fatigue and was subsequently diagnosed with hypercalcemia with an ionized calcium of 1.52 mmol/L (reference 1.15-1.33). His plasma PTH levels were also elevated at 18 pmol/L (reference: 1.6-6.0). The patient was referred to our department for a surgical consult. Clinical examination showed no palpable neck mass. Neck ultrasound identified a 10 mm enlarged right inferior parathyroid gland. No further localization study was performed. The patient was planned for a focused parathyroidectomy, which was performed a month later. The excised gland weighed 700 mg and measured 19 mm. Upon macroscopic examination, the tumor exhibited a cystic appearance. Histologically, the lesion demonstrated a multilobar, cord-like cystic growth pattern, with a single-lined, pseudostratified epithelium built-up by cylindrical cells with basal-positioned nuclei and vacuolated cytoplasm  and polyclonal Paired box 8 (PAX8), thereby verifying them as parathyroid-derived , and he was discharged the same day. Subsequent ionized calcium measurements 3 weeks later showed normalized levels at 1.25 mmol/L, and values remains normal at follow-up, although PTH levels one year postoperatively was just above normal range, probably secondary to a synchronous vitamin D deficiency at the time. The patient is well and without any related symptoms as of May 2021.We report a parathyroid adenoma with a highly unusual growth pattern, characterized by cysts lined by a respiratory-like epithelium. The parathyroid origin was proven by immunohistochemistry, and the functionality of the lesion was also established as surgical removal cured the patient from hypercalcemia. The finding is of potential value for practicing endocrine pathologists as a novel differential diagnosis when investigating cystic lesions lined by a ciliated, cylindric epithelium in the neck.Usually, the finding of cystic structures lined with cylindrical epithelium in the neck could lead the pathologist to suspect a thyroglossal duct cyst . HoweverWhen searching online repositories for similar findings, no credible descriptions of this phenomenon was identified, and the incidence of this type of parathyroid tumor is therefore assumed to be exceedingly low. On an experimental basis, ozone exposure in dogs has been found to prompt the development of ciliated cystic structures within the parathyroid glands , 11. EveTo conclude, we describe a parathyroid adenoma with a previously unreported histological growth pattern. Future retrospective analyses of large tumor series can possibly uncover similar cases in which the cellular features were initially overlooked.The original contributions presented in the study are included in the article/supplementary material. Further inquiries can be directed to the corresponding author.Ethics approval no. 01-133 was obtained by KI Forskningsetikkommitté Nord. The patients/participants provided their written informed consent to participate in this study. Written informed consent was obtained from the individual(s) for the publication of any potentially identifiable images or data included in this article.CCJ conceived the idea, reviewed the histology and drafted the manuscript. JZ reviewed the clinical history and edited the manuscript. All authors contributed to the article and approved the submitted version.CCJ is a Junior Clinical Investigator sponsored by the Swedish Cancer Society.The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.All claims expressed in this article are solely those of the authors and do not necessarily represent those of their affiliated organizations, or those of the publisher, the editors and the reviewers. Any product that may be evaluated in this article, or claim that may be made by its manufacturer, is not guaranteed or endorsed by the publisher.


Pleomorphic xanthoastrocytoma (PXA) is an infrequent neoplasm that affects children less commonly than adults. In this case report, a four-year-old boy presented with focal seizures has diagnosed with this tumor in the frontal lobe. Complete surgical resection was achieved, and histopathological features of PXA grade II were observed. During follow-up, the patient showed improvement of the focal seizures. In spite of the pleomorphic features, the PXA had a favorable prognosis. Pleomorphic xanthoastrocytoma (PXA) is a low-grade glial neoplasm originates from subpial astrocytes and first described at the end of the seventies -3. In adPXA could arise in many locations of the central nervous system (CNS), such as the temporal, parietal, and frontal lobes; cerebellum; pineal region; and spinal cord . AccordiThe goal of this case report was to elucidate the overall characteristics of PXA in order to provide a differential diagnosis and present a literature review.A four-year-old boy first presented with epileptic focal seizures in the right hemiface without generalization for three months. Care of the patient was primarily provided by a neurologist, who initiated carbamazepine therapy, which achieved a satisfactory control of the patient’s condition. In addition, the patient had no compromised physiological milestones and had normal neurocognitive developmental and regular neurological examination results. Brain imaging was performed, and magnetic resonance imaging displayed a tumor in the left frontal lobe which depicted a heterogeneous shape, with solid and cystic portions Figure .Thus, surgical resection was indicated.For the surgical technique, the patient was placed in a prone position, with his head fixed with pins. Neurophysiological intraoperative monitoring was conducted owing to the close proximity of the tumor to the precentral gyri. The somatosensory-evoked potential monitoring and brain mapping were performed during surgery. Left frontoparietal craniotomy was performed, and after the dura was opened and directed toward the superior sagittal sinus, a small corticectomy was performed in the superior frontal sulcus. Macroscopically, the solid part of the tumor had a remarkably thick consistency, and the cystic portion was completely occupied by a translucid fluid. The cyst was surrounded with a thick capsule.As complete resection was achieved, hemostasis was performed, and the bone flap was fixed using absorbable plates and screws. The patient demonstrated an uneventful postoperative period.The tumor sample depicted marked heterogeneity and was formed by ovoid and multinuclear giant cells. In addition, some areas featured cytoplasmic inclusions and high degrees of vascularization. Therefore, the pathology was consistent with PXA grade II Figure .The patient showed adequate control of seizures. At four months after hospital discharge, the anticonvulsant was removed from the treatment regimen. No adjuvant treatment was needed, and the tumor did not recidivate.In this case report, we describe a PXA occupying the frontal lobe in a child who presented with focal seizures. Although cases are infrequent in the young age group, such neoplasm could be diagnosed in a wide range of ages and are responsible for less than one percent of all brain tumors . FurtherThe imaging feature observed in the present case report was a solid mass surrounded by a cystic portion with a mass effect and no hemorrhage signs. The heterogeneous appearance with peripheral contrast enhancement led us to consider other differential diagnoses such as primitive neuroectodermal tumors, pilocytic astrocytoma, atypical teratoid rhabdoid tumors, and glioblastoma ,13. NeveSurgical resection remains the cornerstone of treatment in which combination with a low tumor mitotic rate would improve the overall outcome. In addition, anatomically, this tumor presents more superficially in the brain, allowing for an easier surgical approach . On the Concerning adjuvant therapy, the best evidence does not support the use of adjuvant radiotherapy. In a recent meta-analysis, 17.6% of 167 patients received this treatment, but this proportion was considered insufficient to obtain evidence. Therefore, the authors concluded that both radiotherapy and chemotherapy are not recommended for PXA . HoweverMicroscopically, this lesion presented moderated cellularity, predominantly pleomorphic tumor with no necrosis and rare mitoses, and occasional presence of bizarre multinucleate cells ,17. In sPXA is a rare brain tumor for which maximum operative resection is the mainstay for achieving the best outcome owing to the lack of evidence of the effectiveness of adjuvant radiotherapy and chemotherapy.


Ehlers Danlos syndromes (EDS) are a group of genetic disorders, characterized by skin hyperelasticity, joint hyperlaxity and tissue weakness. Vascular EDS is rare and is differs from other types of EDS by an inconsistent acrogenic morphotype and the occurrence of severe digestive and vascular complications, which can be lifethreatening.We report the case of a 27-year-old man with a type IV vascular Ehlers-Danlos syndrome revealed by a colonic perforation after appendectomy for peritonitis secondary to appendicitis. The etiology of the perforation remained a challenge till a genetic research was carried out for COL3A1 gene mutation, which was positive in favor of vascular Ehlers Danlos disease. Then, a totalization of the colectomy with ileorectal anastomosis was performed.Vascular Ehlers Danlos syndrome (VEDS) is due to qualitative and quantitative abnormalities in the synthesis of type III collagen, which is a major constituent of the vessel wall, skin, joint capsules, uterus and gastrointestinal tract, particularly the colon. Colonic perforation, particularly sigmoidal perforation, is the most frequent complication in SEDV and most often precedes the molecular diagnosis. Colonic perforations are uncommon. The Hartmann procedure is a well-established surgical treatment modality, especially for emergency surgery. Given the iterative risk of colonic perforation and anastomotic leakage, preventive treatment by total colectomy with ileo-rectal anastomosis or definitive ileostomy is recommended by several authors.SEDV is a rare pathology with a difficult diagnosis. However, it should be keeped in mind when there is any spontaneous colonic perforation in the young people. •Ehlers danlos syndromes are a group of genetic disorders of the connective tissue.•Life expectancy is reduced due to spontaneous vascular rupture or colonic perforation.•Colonic perforations are uncommon. They occur in the context of diverticulitis in adults.•The diagnosis is difficult because of the rarity of the pathology and the inaccessibility of genetic tests.•The surgical management of colonic perforations in ehlers danlos syndromes is not codified. Its prevalence is estimated at 1 per 150 000 .Life expectancy is reduced (mean age 45–50 years), due to spontaneous vascular rupture or colonic perforation . The firWe report the case of a 27 year old man with vascular Ehlers Danlos syndrome revealed by sigmoid perforation for whom a total colectomy was performed with deferred ileorectal anastomosis as a preventive measure. The aim of this case is to discuss the place of preventive total colectomy in Ehlers Danlos disease according to literature review. This case have been reported in line with scare guideline 2020 .1.1A 27-year-old man with history of appendectomy and peritonitis due to sigmoid perforation. A segmental sigmoidal resection with Hartmann colostomy were performed. The specimen analysis did not reveal any inflammatory sign of bowel disease.Three months later, the patient presented an occlusive syndrome complicated with stoma and the small bowel necrosis. A 43-cm small bowel resection with end to end anastomosis and 10 cm colonic resection associated with a left iliac fossa colostomy rebuilt were performed. The specimen analysis showed moderate congestive interstitial colitis with the presence of numerous eosinophilic polynuclear cells exceeding 40 PNE/field, which could be in favor of eosinophilic colitis, without granulomatous lesions or other signs of IBD. Examination of the bowel resection showed a regular moderately inflammatory mucosa without cryptic abnormalities with some giant cells and rare granulomas in the muscular.-Thin, loose skin,-Hypoplasia of the ear lobule [A].-Madonna face appearance: pinched nose, horizontal lips, prominent cheekbones, and sunken and dark circled eyes [B].-Acrogeria of the hands [C].On third hospitalization, physical examination showed :-Thin, l-Atrophic aspect of the median laparotomy scar-Postoperative eventration of the linea alba with a ring sac 10 cm in diameter.Abdominal examination showed:The remain of physical examination was normal.The etiology of the perforation remained a challenge for this patient and after morphotype analysis, a genetic research was carried out for COL3A1 gene mutation, which was positive in favor of vascular Ehlers Danlos disease. Then, a totalization of the colectomy with ileorectal anastomosis was performed , associa2(5), . Onl. Onl8]. By the way, any idiopathic colonic perforation in a young adult, in the absence of colonic pathology, should require a genetic study for SEDV . In 47% In the literature, the surgical management of colonic perforations in SEDV is not codified, the main reasons being the rarity of the disease, the absence of a formal diagnosis at the time of the first colonic perforation, and the need for emergency surgical treatment ,11.The Hartmann procedure is a well-established surgical treatment modality, especially for emergency surgery. Given the iterative risk of colonic perforation and anastomotic leakage, preventive treatment by total colectomy with ileo-rectal anastomosis or definitive ileostomy is recommended by several authors . HoweverA recently published meta-analysis by Speacke et al. describes 109 operations performed in 51 patients with EDS reported in 44 case series and allows to establish a management strategy in EDS by evaluating the risk of intestinal perforation recidivisme in the therapeutic management. Indeed, the rate of recidivisme is significantly higher after colo-colic anastomosis, compared to ileo-rectal anastomosis , survival analysis also increases with total colectomy compared to other operative modes. The author therefore suggests that total colectomy with ileorectal anastomosis or definitive ileostomy should be performed to prevent the subsequent risk of perforation in any patient with confirmed Ehlers Danlos syndrome.This strategy would limit the risk of certain immediate complications , which has a high (66%).For our patient, a total colectomy with ileo-rectal anastomosis was performed.3SEDV is a rare pathology, the diagnosis is difficult because of the ignorance of the pathology and the inaccessibility of genetic tests. However, it should be keeped in mind when there is any spontaneous colonic perforation in the young people.The surgical management of colonic perforations recommends total colectomy with ileo-rectal anastomosis or definitive ileostomy as a preventive measure, justified by the high rate of recurrence and anastomotic fistulas.The study is exempt for ethical approval.None.Khalid Elhattabi: manage the research and design and write the first draft of the manuscript.Hasna Benghait: manage the research and design and write the first draft of the manuscript.Abdelilah Elbakouri: manage the research and design and write the first draft of the manuscript.Mounir Bouali: manage the research and design and write the first draft of the manuscript.Fatimazahra Bensardi: correction of the paper.Abdelaziz Fadil: correction of the paper.This is not the first case and the registration is not required.The Guarantor is the one or more people who accept full responsibility for the work and/or the conduct of the study, had access to the data, and controlled the decision to publish.The authors declare having no conflicts of interest for this article.


An unicornuate uterus with a non-communicating rudimentary horn (UUNCRH) is a very rare uterine malformation which is difficult to diagnose and to decide the surgical plan. We aim to describe the case of pediatric UUNCRH patient and our operative technique of laparoscopic intra-uterus indigo carmine injection (LIUICI) to confirm that the rudimentary horn (RH) is non-communicating before the resection and review the relevant literature to ascertain the most appropriate treatment option in these patients.A 11-year-old girl who developed progressive severe abdominal pain and dysmenorrhea was referred to our hospital. Uterine malformation and right hematosalpinx was confirmed with magnetic resonance imaging (MRI). Pre-operative treatment with a gonadotropin-releasing hormone agonist enabled improvement in the symptoms. Laparoscopic exploration was scheduled. The right fallopian tube was resected laparoscopically and a 3Fr tube was inserted into its cut end. Indigo carmine injected in the RH through the tube. No leakage of indigo carmine was found from the vagina, indicating the diagnosis of the uterine malformation is an UUNCRH and we performed the resection of the RH safely.In pediatric patients transvaginal detailed examination is not easy to perform. Therefore, diagnostic and operative laparoscopy is critically important for the safe treatment. In addition, laparoscopic removal of a RH can be used to decrease the incidence of adhesions.We found LIUICI technique before the resection of the RH is safe, technically feasible and minimally invasive approach for pediatric UUNCRH patients. A unicoUU with a non-communicating RH (UUNCRH) patients may present with dysmenorrhea, chronic pelvic pain and hematometra . In the In recent years, laparoscopy has become a viable alternative to laparotomy for management of the RH. However, it is difficult to ascertain preoperatively if the RH is communicating and to reveal the detail of the uterus anomaly in children, because transvaginal examination for young children is limited to perform in the outpatient clinic. Therefore, proper intra-operative diagnosis and surgical treatment are essential to prevent serious complications.We aim to describe our operative technique of laparoscopic intra-uterus indigo carmine injection (LIUICI) to confirm that the rudimentary horn is non-communicating before the resection and review the relevant literature to ascertain the most appropriate treatment option in these UUNCRH patients.2We ensure our case report is compliant with the SCARE Guidelines 2020 .2.1A 11-year-old girl presented severe pain during her menstruation period which was only minimally relieved with medical management, non-steroidal anti-inflammatory drugs (NSAIDs). Uterine malformation and right hematosalpinx was confirmed with magnetic resonance imaging (MRI) ab. Pre-o2.2During the operation, a patient was placed in a lithotomy position under general anesthesia. First, a 20 mm single vertical umbilical incision was made. The Lap-protector  was then placed through the incision. After creating a pneumoperitoneum of 6–10 mmHg, a 5 mm, 30 degree laparoscope was inserted and three 5 mm trocars  were placed both sides of lower abdomen and left side of upper abdomen. Then, laparoscopic exploration confirmed the presence of uterine malformation  and righ3The incidence of congenital Mullerian duct anomalies has been reported as high as 3.2% ,10. A paThe UU with RH seems difficult to diagnose. The RH is often small and is not easy to palpate during bimanual examination . The ultWe report a case of a UU with a non-communicating RH presenting as severe dysmenorrhea. In our case, laparoscopic exploration confirmed the right hematosalpinx and the right fallopian tube was resected laparoscopically. Then, we inserted 3Fr feeding tube into the RH and injected indigo carmine. We confirmed that the diagnosis of the uterine malformation is an UUNCRH. Owing to this procedure, we performed the resection of the RH safely. Furthermore, the scar was very small and the patients had good cosmetic results.4The LIUICI technique was valuable to exclude a communicating uterine horn before minimal-invasive resection in patients with UUNCRH. The technique is simple and safe to perform laparoscopically.The authors declare that they have no competing interests.The authors declare that they received no funding support for this study.Not applicable.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.TT acquired the data and drafted the manuscript. TT, SS, HK and SY performed the operations. All other authors attended the patient postoperatively. All authors read and approved the final manuscript.Not applicable.Not applicable.Not commissioned, externally peer-reviewed.


Urge incontinence is the most frequent type of urinary incontinence that can be due to bladder outlet obstruction or overactive bladder. A focused history is crucial to identify the type of urinary incontinence and the possible etiology. We report the case of a 67-year-old man with urinary frequency, urgency, and nocturia. However, his urine stream is normal with no history of an intermittent stream, hesitancy, or postvoid dribbling. Digital rectal examination revealed normal prostatic size. Urinalysis results were normal. Urine culture showed no growth. The patient was prescribed symptomatic treatment in the form of anticholinergic medication but failed to provide any clinical improvement. Urodynamic studies suggested the diagnosis of detrusor instability. A computed tomography (CT) scan of the abdomen was performed and demonstrated the presence of a large retroperitoneal lipoma exerting a mass effect on the bladder. The mass was successfully resected by laparotomy operation. Following the operation, the patient had complete resolution of his symptoms. The retroperitoneal region is an extremely rare site for lipoma. Patients with urinary urgency should be carefully evaluated for any structural lesion causing a compressive effect on the bladder. Urinary incontinence, the involuntary leakage of urine, may result from different etiologies. The types of urinary incontinence include urge urinary incontinence, stress urinary incontinence, and mixed urinary incontinence . In men,We present the case of a 67-year-old man who presented to the outpatient clinic with urinary frequency, and urgency, and incontinence. He reported having these symptoms for the last one year. Recently, his symptoms worsened and he needed to void every two hours and had nocturia multiple times a day. There was no history of difficulty with voiding, poor or intermittent urine stream, or postvoid dribbling.His past medical history is remarkable for long-standing hypertension, diabetes mellitus, and dyslipidemia. He has been on amlodipine 5 mg once a day, metformin 1000 mg three times a day, and atorvastatin 20 mg once a day. He did not undergo any previous surgical operations. He worked as an accountant. He had smoked 10 cigarettes daily for the last 20 years. He consumed alcohol occasionally. The family history was non-contributory.Upon examination, the abdomen was soft with no tenderness or palpable masses. Digital rectal examination revealed no enlargement of the prostate. Examination of other systems was normal. The prostate-specific antigen level was 2.1 ng/ml. Urinalysis findings were normal. Urine culture revealed no significant growth. Basic laboratory investigations were within the normal limits (Table In the subsequent visit, the patient reported progression of his symptoms and he started to have urge incontinence. The urodynamic analysis confirmed the presence of involuntary contractions of the detrusor muscles during the filling phase representing detrusor instability and overactive bladder. An abdominal CT scan was performed to rule out any structural abnormalities as an etiology of the overactive bladder.The CT scan demonstrated a large retroperitoneal circumscribed low attenuation lesion consistent with lipoma. The lesion was extending into the pelvic cavity and exerting a pressure effect on the bladder Figures , 2. SubsWe reported the case of a retroperitoneal lipoma causing detrusor instability. Lipoma, a benign tumor of mature adipose tissue, is the most common soft tissue tumor in adults. However, the development of lipoma in the retroperitoneal region is exceedingly rare. Further, retroperitoneal tumors are rare and account for <0.5% of all neoplasms in the body. Notably, Weniger et al.  have conThe exact pathogenesis of lipoma remains unclear. Obesity and dyslipidemia have been linked to subcutaneous lipomatous lesions. However, the available data does not suggest that these are risk factors for retroperitoneal lipoma . The majA computed tomography scan can make the diagnosis of retroperitoneal lipoma very accurately. Retroperitoneal lipoma exhibits the general features of a lipomatous lesion with an encapsulated lesion of fatty attenuation that has no soft tissue component . HoweverThe retroperitoneal region is an extremely rare site for lipoma. Patients with urinary urgency should be carefully evaluated for any structural lesion causing a compressive effect on the bladder. A CT scan is the investigation of choice to make the diagnosis of retroperitoneal lipoma. Surgical resection is recommended since deep lipomatous lesions are often malignant.


Abdominal cocoon (AC) or Encapsulating Peritoneal Sclerosis (EPS) is a rare cause of bowel obstruction and due to non-specific presentation, it can be misdiagnosed and often mistreated.We present the case of 42 years male with a history suggestive of complete small bowel obstruction (SBO) without a history of pulmonary tuberculosis (TB) or peritoneal dialysis. CT imaging as well as the intraoperative finding of a cocoon membrane encasing the small bowel led to the diagnosis of abdominal cocoon.Abdominal cocoon can be idiopathic or secondary to peritoneal dialysis, tuberculosis, or other rare causes. Patients usually present with features of SBO with varying severity. Diagnosis is aided by imaging investigations mainly CT scan and management is primarily surgical and usually involves adhesiolysis, total removal of the membrane with or without bowel loop resection.Diagnosis of abdominal cocoon warrants awareness of the disease and a high index of suspicion of the treating clinician in patients with intestinal obstruction and an abdominal lump without a history of previous abdominal surgery. CT can guide diagnosis and early operative management seems to bear the best outcomes. •Abdominal cocoon is a rare cause of intestinal obstruction with high morbidity and mortality if not diagnosed in time.•We report a case of an abdominal cocoon in an adult male without any obvious preceding cause.•Diagnosis warrants a high index of suspicion in patients with features of intestinal obstruction and an abdominal lump.•CT can guide diagnosis and early operative management seems to bear the best outcomes. EPS may be idiopathic or associated with peritoneal dialysis, TB infection of the abdomen, and other rare causes.We present a case of complete small bowel obstruction due to cocoon formation who underwent operative management. This case has been reported in line with the SCARE checklist 2We present the case of 42-year-old male with a history of right upper quadrant abdominal pain, vomiting, and inability to pass stool and flatus for two days. The pain was acute in onset, severe, and associated with four episodes of non-bilious vomiting. With these symptoms, he noticed a non-tender, firm lump palpable in the right upper quadrant. There was no past history of pulmonary TB or peritoneal dialysis.He had a similar episode one month back which was managed conservatively.Per abdominal examination revealed a soft non-distended abdomen with a visible intraperitoneal mass about 4 × 5 cm over the right lumbar region. Bowel sounds were exaggerated.9/l, neutrophils 65%, lymphocytes 30%) with rest parameters being normal. Plain radiographs of the abdomen showed multiple air-fluid levels with dilated small bowel loops. Chest radiograph was unremarkable and sputum Acid Fast Bacilli stain reported negative. TB was not detected in Gene-Xpert of sputum. Ultrasonography of the abdomen and pelvis showed dilated bowel loops but characteristic trilaminar membrane was not noted.Laboratory investigations were sent which showed leukocytosis , Romi Dahal (RD), Deepak Sharma (DS), Rupesh Verma (RV), Gyaneswor Shrestha (GS) = Study concept, Data collection, and Surgical therapy for the patientOm Prakash Bhatta (OPB), RV, GS = Writing - original draft preparation and editingPBK, RD, DS = Senior author and Manuscript reviewer.All authors critically reviewed, revised and contributed to the final article. All authors read and approved the final manuscript.Nothing to declare.


Ectopic breast tissue is the presence of retained breast tissue along the embryologic mammary ridge, also known as the milk line. Accessory tissue can be located anywhere along or outside the anatomic milk line extending from the axilla to the groin. Ectopic breast tissue can undergo the same physiologic and pathologic changes seen in normal breast tissue, such as fibroadenomas, fibrocystic changes, and malignancy. The wide range of clinical presentations and symptomatology can pose a significant diagnostic challenge, and clinicopathologic correlation is key in establishing the diagnosis. In this report, we review the clinical and histopathologic findings in a rare case of cutaneous periumbilical fibroadenomas in a 25-year-old female. Ectopic fibroadenoma is a benign tumor of the breast that is most commonly found in the axilla but can occur in any location along the anatomic distribution of the embryologic milk line -3. LesioIn this case report, we discuss a young woman who presented with a three-year history of progressively enlarging periumbilical growths. Excision of these nodules demonstrated glandular epithelial and stromal hyperplasia confirming the diagnosis of ectopic fibroadenomas.A 25-year-old African-American female presented with a three-year history of enlarging growths in the periumbilical region. The growths originally began as small subtle bumps under the skin and slowly enlarged over time. They were not tender to palpation but were occasionally painful with movement and during menstrual cycles. Physical examination revealed two flesh-colored, ~2.0 x 2.5 cm rubbery, non-tender, mobile, well-circumscribed and non-reducible nodules that appeared confined to the subcutaneous tissue Figure . The remExcisional biopsy of a periumbilical lesion revealed a well-circumscribed neoplasm comprised of stromal fibrosis and glandular epithelial hyperplasia in a pericanalicular to primarily intracanalicular pattern, with no evidence of stromal or epithelial dysplasia/atypia Figure .The patient's histopathological and clinical findings were most consistent with a diagnosis of ectopic cutaneous fibroadenomas. During embryonic development, breast tissue and glands grow along mammary ridges . MammaryEctopic breast tissue can undergo physiologic and pathologic changes seen in normal breast tissue such as mastitis, fibrocystic changes, and fibroadenoma ,2. FibroThe differential diagnosis, in this case, includes fibroadenoma, lipoma, cyst, and although less likely a cutaneous metastasis from an underlying malignancy . DependiThis report demonstrates a unique case of ectopic fibroadenoma diagnosed in a patient with multiple growths in the periumbilical region. Histopathology in addition to clinical presentation helped to establish the diagnosis of fibroadenomas in this patient. Both lesions were completely removed via excision with no evidence of recurrence to date.Ectopic fibroadenomas are rare benign tumors containing both fibrous and glandular tissues. They can occur anywhere along the milk line but are seen most commonly in the axilla. Ectopic fibroadenomas can be asymptomatic or produce a variety of hormone-sensitive symptoms such as tenderness, irritation, erythema, and milk production. Ultrasound, mammography, or MRI can be used for initial evaluation. However, characteristic histopathologic evaluation is required for definitive diagnosis. Clinicopathologic correlation is critical in establishing the diagnosis, stratifying risk for malignant transformation, and guiding treatment for this obscure disease entity.


Primary ovarian high-grade endometrial stromal sarcoma is a very rare disease. Even though it has poor prognosis, the gold standard treatment has not been established owing to its rarity. This report aimed to present therapeutic options for primary ovarian high-grade endometrial stromal sarcoma.2). We performed the secondary debulking operation including total hysterectomy, metastasectomy, omentectomy, peritonectomy, appendectomy, and hyperthermic intraperitoneal chemotherapy (paclitaxel 175 mg/m2). Currently she has been alive for 28 months under a new chemotherapy regimen.A 49-year-old Asian woman presented with disseminated intravascular coagulation due to ruptured primary high-grade ovarian endometrial stromal sarcoma with multiple intraperitoneal metastases. After the initial surgery, the patient underwent adjuvant chemotherapy with three courses of Adriamycin (75 mg/mWe suggest cytoreductive surgery with hyperthermic intraperitoneal chemotherapy could be a therapeutic option for primary high-grade ovarian endometrial stromal sarcoma with peritoneal dissemination. Endometrial stromal sarcoma (ESS) is a rare malignant tumor of the uterus, which accounts for 0.2% of all uterine malignancies and less than 10% of the all uterine sarcomas . The mesWhile low-grade ESS shows an indolent clinical course and a relatively good prognosis, high-grade ESS has poor prognosis due to rapid progression of the disease and high incidence of recurrence and metastasis. Treatment includes surgery and adjuvant therapies including chemotherapy and radiation therapy, but the gold standard has not been determined because of the limited number of cases , 7.Here we describe a case of ruptured primary high-grade ovarian ESS with multiple intraperitoneal metastases being treated with cytoreductive debulking surgery and hyperthermic intraperitoneal chemotherapy (HIPEC) followed by adjuvant chemotherapy.3/μL], C-reactive protein (CRP) elevation of 25.8 mg/dL with prolongation of prothrombin time (PT) (90.4 seconds), PT international normalized ratio (INR) [A 49-year-old Asian woman presented with severe abdominal distension and dyspnea. She suffered from the feeling of swelling abdomen, dull nature abdominal discomfort and pain, edema in both legs, and dyspnea from 3 weeks before her visit. She visited another hospital 2 days prior to her visit and was transferred to our emergency room owing to huge ovarian cystic mass occupying almost all of the abdominal cavity and large amount of left pleural effusion on computed tomography (CT) scan. On initial vital sign assessment, her blood pressure was stable and she had tachycardia but no fever. Her laboratory findings showed anemia (hemoglobin 8.3 g/dL), leukocytosis [white blood cells (WBC) 21.00 × 102). On the follow-up CT scan that was performed 6 months after the chemotherapy, new 11-mm-sized probable seeding nodules in the right omentum and left paracolic gutter were seen. A 1.5-cm-sized partly solid nodule in the right upper lung field was also identified on the chest CT. As for the lung lesion, we had both primary and metastatic lesion in mind, and planned to perform the surgery separately.On the initial operation findings, ruptured 30-cm-sized right ovarian multiseptated cystic mass showing diffuse adhesion to retroperitoneum, abdominal wall, and uterus was identified. Although frozen section biopsy from the right ovarian mass was revealed as a poorly differentiated carcinoma, debulking surgery including hysterectomy could not be performed owing to persistent oozing pattern bleeding from multiple sites, which was due to DIC. We performed bilateral salpingo-oophorectomy and partial omentectomy. The pathologic diagnosis of the right ovary was high-grade stromal sarcoma with features of brisk mitosis (30–40/high-power field), focal necrosis, adenofibromatous component, endometriosis with mucinous metaplasia, and hypercellular stroma Fig. . The tum2). In the final pathologic report, it was confirmed that the uterine cervix, myometrium, and endometrium had no specific finding except atrophy, which excludes the possibility of endometrial origin malignancy. Peritoneum, omentum, and appendix specimens were confirmed as metastatic high-grade endometrial stromal sarcoma. Two months after the surgery, the patient underwent another right upper lung lobectomy operation, and the biopsy was revealed as adenocarcinoma, which means double primary malignancy. Currently, she has been alive for 28 months under a new chemotherapy regimen: paclitaxel (175 mg/m2) and ifosfamide (1.6 g/m2).We performed the secondary debulking operation including total hysterectomy, metastasectomy, omentectomy, peritonectomy, appendectomy, and HIPEC , low-grade endometrial stromal sarcoma (LG-ESS), high-grade endometrial stromal sarcoma (HG-ESS), undifferentiated uterine sarcoma (UUS), and uterine tumor resembling ovarian sex cord tumor (UTROSCT) . Regardip = 0.007) [The standard treatment of ESS confined to the uterus is total extrafascial hysterectomy with or without bilateral salpingo-oophorectomy, but the treatment of extrauterine ESS is controversial. One retr= 0.007) . Systemi= 0.007) There have been several retrospective reviews about the possibility of cytoreductive surgery followed by hyperthermic intraperitoneal chemotherapy (HIPEC) to be a promising treatment modality in patients with high-grade uterine sarcoma with peritoneal dissemination -16. MultPrimary ovarian high-grade endometrial stromal sarcoma (ESS) is a very rare disease. Even though it has poor prognosis, the gold standard treatment has not been established owing to its rarity. This report aimed to present therapeutic options for primary ovarian high-grade ESS. We suggest cytoreductive surgery with HIPEC could be a therapeutic option for high-grade uterine sarcomas with peritoneal sarcomatosis as well as for primary high-grade ovarian ESS with peritoneal dissemination. Although large studies might be challenging owing to the rarity of the disease, further study is needed.