Haemangioma or hemangioendothelioma is amongst the commonest developmental, vascular lesions of infancy and childhood. Hemangioendothelioma of the salivary glands, however, is extremely rare. Due to their rarity, they may be misdiagnosed as lymphangiomas or other cystic lesions found more commonly in the region. This may lead to surgical complications including torrential hemorrhage that may have dire consequences for the patient. Herein we present the case of a nine-year-old male with a cavernous haemangioma involving the left submandibular gland which was diagnosed on-table due to inconclusive pre-operative radiological and pathological diagnosis. Fortunately, due to meticulous dissection and care the lesion was excised in toto without any significant blood loss. Haemangioma of the submandibular gland is so uncommon that often it isn't even considered a differential diagnosis for cystic swellings and lesions in this region. Mistaken diagnoses preoperatively may prove disastrous for the patient. Excision of haemangiomas requires planning for hemostasis and blood loss if it occurs. Even minor iatrogenic manipulation of vascular lesions may completely obscure the field due to bleeding, making dissection and recognition of anatomical landmarks very difficult. This is especially dangerous in the submandibular region due to the proximity of various vital vascular and neural elements. A differential of haemangioma should therefore always be considered by surgeons and radiologists alike for lesions with suspicious or indeterminate features, in this region.  Haemangiomas are amongst the commonest benign lesions. However, salivary gland haemangiomas are very rare ,2. UnlesA nine-year-old male presented to the ENT OPD with the chief complaint of a left-sided neck swelling of three months duration. It was associated with facial disfigurement and restricted neck movements. The informant (father) revealed that the swelling was insidious in onset and gradually increased in size. Initially, two pea-sized swellings were observed which then coalesced and grew to attain the current size. It was painless and not associated with fever, weight loss, or malaise. There was no history of difficulty swallowing or breathing. There were no relevant co-morbidities. It was diagnosed as a case of lymphangioma after contrast-enhanced CT of the neck at a hospital elsewhere two months previously. Two doses of intra-lesional bleomycin (3IU/mL) were administered six weeks apart. After the first dose, the swelling reduced in size. After the second dose, however, the swelling increased in size and the consistency changed from soft to firm. FNAC was repeated at the same hospital which was then suggestive of hematoma. On presentation at our hospital, local examination revealed a solitary, 6*5 cms ovoid, firm, mobile, non-compressible, non-reducible, non-tender mass extending from the left angle of the mandible laterally to midline medially. It extended up to the hyoid bone inferiorly, overlying the ramus of the mandible, and occupied the entire submandibular region . The margins were regular. The transillumination test was negative. The overlying skin and local temperature were normal. On oral cavity examination, the left tonsillar region was pushed medially. The floor of the mouth was found to be elevated. The lesion was bimanually palpable. There was no evidence of cervical lymphadenopathy.Routine blood investigations were within normal limits. The Mantoux test was negative.Contrast-enhanced CT of the neck revealed a large, lobulated, multi-septate, cystic lesion in the left submandibular gland extending into the left para-pharyngeal space. Superiorly it extended up to the nasopharynx, the left parotid region, and masseter muscle and inferiorly to the sublingual space .There was evident compression of the oropharyngeal airway with rightward displacement. There was no calcification or underlying bone changes. The patient was taken up for surgery after appropriate pre-anesthetic clearance and written and informed consent. Under general anesthesia with orotracheal intubation, the subcutaneous tissue, fat, and fascia over the lesion were carefully dissected. .The marginal mandibular nerve was found to be splayed and partially embedded in the capsule of the lesion. It was carefully separated and retracted superiorly. During the dissection one locule of the lesion burst and dark red, fresh blood spilled out, leading us to suspect that this may in fact be a vascular lesion. The capsule extended from the skull base to the hyoid bone and was carefully separated by blunt and sharp dissection. The facial artery, facial vein, and submental artery were identified and ligated. The hypoglossal nerve and lingual nerve were identified and preserved. The lesion was excised in toto and sent for histopathological examination. The cumulative blood loss during the procedure was around 200 ml. Histopathological examination showed a single globular tissue, measuring 6*6*3 cm with multiple cysts. On sectioning, it yielded hemorrhagic fluid and coagulated blood.Microscopic examination showed cystically dilated, endothelium-lined blood vessels filled with intact and lysed red blood cells and fibrin deposition. Surrounding tissue had few thin-walled blood vessels, dense lymphoplasmacytic infiltrate, skeletal muscle bundles, and normal salivary gland tissue with areas of hemorrhage. These features were diagnostic of cavernous hemangioma of the submandibular gland .Post-operatively, the patient was asymptomatic with minimal serosanguinous discharge in the drain over 24 hours. Intravenous antibiotics and analgesics were administered. There were no features of facial nerve palsy or any other complications. The patient was discharged on postoperative day seven. On follow-up visits at one week and six months, the patient was asymptomatic with complete relief from the pre-operative symptoms.Hemangiomas are vascular abnormalities that are characterized by increased proliferation and renewal of endothelial cells . HaemangIt encompasses three distinct clinical entities - capillary hemangioma, cavernous hemangioma, and mixed hemangioma. Compared to capillary hemangioma, cavernous hemangioma is rare and tends to occur in older children and adults . Also unThe parotid gland is the most common salivary gland involved. Childers et al found only ten cases of salivary gland haemangiomas in a 30-year retrospective study of all cases of haemangiomas retrieved from the Registry of Oral and Maxillofacial Pathology of the Armed Forces Institute of Pathology at Washington DC, all of which were located in the parotid gland . In anot Hemangioma of the submandibular gland is extremely rare. Our PubMed and Medline search of English language reports revealed only 17 cases of submandibular haemangioma. Of these, eleven cases occurred in females and six cases were seen in males.  The patients ranged from 7–65 years and the average was 36 years (Reports where the gland was completely absent, as in the cases reported by Hopkins et al and Iguchi et al, are not included amongst these. Also, submandibular gland haemangioma extending to para-pharyngeal space is even more unusual with only one previously reported case with similar manifestations . Ours isVenous haemangioma and arterio-venous malformations have also been reported in the submandibular gland. However, very few cases have been reported so far of such venous malformations -23.90% of all haemangiomas are seen in children and young adults . Nagao eMost of the infantile haemangiomas in salivary glands are capillary haemangiomas. Only the cavernous variant is generally found in adults. One case of submandibular cavernous haemangioma has been reported previously in a young child . In thisRecurrent inflammation may also lead to mass being firm on palpation, due to subsequent fibrosis . EpisodiStudies have shown that the specificity and sensitivity of 99mTc scintigraphy are as high as 100% and 89% respectively for cavernous haemangioma . These d FNAC is often inconclusive and reveals only blood components presenting as a hematoma. In fact, in all the reported cases of submandibular haemangioma, the FNAC was inconclusive. An indeterminate FNAC with radiological features of a vascular pathology should raise the suspicion of a haemangioma. If a vascular lesion is suspected early on, FNAC should be avoided to prevent iatrogenic trauma and hematoma formation. Because cavernous hemangiomas tend to be larger, less well-circumscribed than capillary hemangiomas, and show no tendency to regress, surgery remains the management modality of choice. In all the previously reported cases the lesions were surgically excised in toto with the submandibular gland. Other treatment approaches that may be attempted include laser surgery, cryosurgery, and injection of sclerosants, ligation of the feeding vessel, embolization, and corticosteroids. Due to the infrequency of its incidence, a high degree of suspicion is imperative for accurate diagnosis and prompt management. A differential diagnosis of vascular malformations should be considered for all soft, cystic lesions in the head and neck region and evaluated accordingly. Inaccurate and inadequate pre-operative evaluation of such lesions may prove catastrophic for the patient. The most commonly encountered cystic lesions in this region are usually lymphangiomas. The pre-operative groundwork and surgical approach to lymphangioma and haemangioendothelioma differ radically. Haemangioma excision requires meticulous planning for hemostasis and blood loss. Also, intra-operatively the dissection needs to be cautious and meticulous to avoid intractable hemorrhage, as it may obscure the field making dissection and recognition of anatomical landmarks very difficult. This is especially dangerous in the submandibular region due to the proximity of various vital vascular and neural elements. Cho et al observed that torrential hemorrhage occurred in patients in whom the surgeons were unable to ligate the feeding vessel . Aynali In conclusion, a differential diagnosis of haemangioma should always be considered by surgeons and radiologists alike for lesions with suspicious or indeterminate features, in this region.


Rhabdomyosarcoma (RMS) is a soft tissue tumor, which is the most common in the head, neck, limbs, and trunk. RMS originating from the epididymis is extremely rare. Herein, we reported a 34-year-old patient with RMS on the right epididymis. For this case, right epididymal mass resection was performed and intraoperative freezing suggested a malignant tumor. Right testicular radical resection was subsequently adopted, with right epididymal alveolar RMS being pathologically diagnosed. Alternating VAC/VI chemotherapy was given after surgery, and tumor recurrence has not been found so far. Rhabdomyosarcoma (RMS) is an extremely rare solid malignant tumor, it tends to metastasize early and is difficult to differentiate from other epididymal tumors. In general, RMS accounts for 5%-10% of childhood tumors, while only 7% originate from epididymis  touched in the right epididymis. The right scrotum was palpated with slight pain, its mobility was poor, but the surface was smooth. An enhanced CT scan observed a tumor in the dorsal lower part of the testicle (3.3×3.0 cm), with heterogeneous enhancement found Figure 1Scrotal RMS primarily originates from particular tissues in children and adolescents. Early diagnosis is critical given its aggressive nature and the relationship between tumor staging and survival. Unilateral scrotal painless swelling is the typical symptom like this patient, while the swelling pain always comes from accompanied epididymitis . UltrasoRMS is a mesenchymal carcinoma which arises from skeletal muscle cells . The mosThe eRMS management is multimodal. Complete surgical resection followed by adjuvant chemotherapy is currently considered to be more appropriate . RecommeRMS is a chemotherapy-sensitive tumor with VAC regimens (vincristine+adriamycin+cyclophosphamide) being the most common regimen to improve survival and reduce recurrence , 13. ForThe main factors affecting the prognosis of eRMS are age, tumor size, pathological type, local recurrence, metastasis, and chemotherapy , 17. ItsIn summary, eRMS is rare and rapidly progressing. Its clinical symptom and imaging manifestation are not specific. So, it is often confused with epididymitis, epididymal tuberculosis, and epididymal tumor, which finally leads misdiagnosis and delays treatment. Patients with scrotal painless swelling, testicular hydrocele, or rapidly growing scrotal mass who is poorly respond to antibiotic should be considered with eRMS. Radical orchiectomy and adjuvant chemotherapy can reduce recurrence and improve survival. Finally, the patient should be monitored for a long time considering RMS is easy to metastasise.This study was funded by National Natural Science Foundation of China (Number: 82060462), Science and Technology Plan project of Guizhou Province (Number: [2019]5405), and the Doctoral Foundation of Guizhou Provincial People’s Hospital (GZSYBS[2018]02). The funding agencies and donors had no role in any aspect of this study.The raw data supporting the conclusions of this article will be made available by the authors, without undue reservation.XL, SW and LL obtained and analyzed the clinical data. XL, SW and TL wrote the manuscript. KJ and JH designed and constructed the figures. KJ, QW designed the study, study supervision and edited the manuscript. All authors contributed to writing and revising the manuscript and figures. All authors contributed to the article and approved the submitted version.This study was also funded by Science and Technology Support Plan of Guizhou Province in 2020 (Fund No. : Guizhou Science and Technology Support [2020]4Y142).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.


Some case series described the use of a combined antegrade and retrograde dilation (CARD) technique to treat complete esophageal stenosis.5Head and neck cancer is a common malignancy with almost 450,000 newly diagnosed cases worldwide each year.In this report, we describe a novel treatment method of radiation-induced complete esophageal stenosis that was treated with combined antegrade-retrograde dilation with the use of needle knife for septotomy.This is a case of a 76-year-old man who was recently diagnosed with oropharyngeal squamous cell carcinoma at the base of the tongue, with metastatic disease to a submandibular lymph node-stage 3 . He was treated with concurrent weekly chemoradiation therapy. He was started on tube feeds through percutaneous endoscopic gastrostomy (PEG) tube at the time of diagnosis because of decreased oral intake and fears of aspiration. The patient continued using the PEG tube throughout the treatment duration for almost 2 months. Three weeks after the last radiation session, he presented with symptoms suggestive of esophageal dysphasia. He was evaluated by a video swallow study that showed aspiration with thin consistency and regurgitation in the proximal esophagus with findings suggestive of luminal narrowing because of soft tissue edematous changes. The patient was therefore scheduled for an endoscopic intervention.The adult esophagogastroduodenoscopy (EGD) was attempted, which showed a benign-appearing severe stenosis 21 cm from the incisors. There was no pinhole, and the scope could not pass Figure . The sep6 The management of complete esophageal stenosis differs from partial stenosis where the antegrade dilation approach is considered the mainstay treatment. The straight antegrade dilation cannot be used with complete luminal occlusion and inability to traverse the stenosis. To overcome this issue, many case series started to use the CARD technique as a treatment for complete esophageal stenosis, providing an alternative to surgical interventions. Those case series showed the CARD procedure was associated with a high success rate, low risk of complications, and improved quality of life. Complications described in those series were mainly in the form of dehiscence of the gastrostomy tract, pneumomediastinum, and perforation.5Complete esophageal stenosis after radiation therapy is not uncommon and is considered a serious adverse event that results in significant morbidity. Before the endoscopic era, management was mainly surgical and sometimes required esophagectomy, which carries high rates of mortality.7 Other techniques that were described in the past included counterpressure forceps, blunt dissection, and direct puncture.8 However, we used the needle knife instead to do the septotomy under direct visualization and transillumination, increasing the success rate and reducing the risk of complications. To our knowledge, this is a very rare case where the CARD technique was used with the use of needle knife septotomy to treat complete benign esophageal stenosis. This technique is probably safe, effective, and minimally invasive and provides hope to be used as an alternative for traditional surgical techniques.After antegrade dilation failed in our patient, we attempted the CARD technique. The introduction of the gastroscopes at both ends of the stenosis provided a good visibility during the whole procedure. We used the adult size gastroscope for the antegrade approach and the ultraslim gastroscope for the retrograde approach. This minimized the risks of gastrostomy tract dehiscence and allowed for an easier access to the esophagus from below the stenosis. In the previous cases where the CARD technique was used to treat complete esophageal obstruction, a glidewire was used most of the time to pull the stenotic tissue proximally, making a bulging tissue which is then grasped and removed with a biopsy forceps.Author contributions: Conception and design: W. Sayeh. Drafting of the article: W. Sayeh, J. Burlen, and S. Ghazaleh. Critical revision of the article for important intellectual content: All authors. Study supervision: A. Tsang, M. Heif, and T. Alhmoud. Final approval of the article: All authors. W. Sayeh is the article guarantor.Financial disclosure: None to report.Informed consent was obtained for this case report.


A weakening or defect in the posterolateral abdominal wall can lead to development of lumbar hernia. These defects are particularly common in the Petit's inferior triangle or the Grynfeltt-Lesshaft superior triangle. There are very few cases of primary lumbar hernias that have been described in the literature till date. As it is a rare entity, it is often misdiagnosed, leading to delay in management. We present a case of a 66-year-old male with no previous surgery who presented with a mass in the left lumbar region for last ten years. The mass gradually increased in size and caused vague dragging pain. On Computed tomography, the diagnosis of Grynfeltt hernia was made. The patient underwent a laparoscopic mesh repair and had an uneventful postoperative hospital stay. Although a rare entity, there should be a high degree of suspicion of a lumbar hernia when evaluating a case of a lumbar mass. Early diagnosis by computed tomography and management with open or minimally invasive techniques can prevent complications. Hafner stated that surgeons would get only one opportunity in their lifetime to diagnose and operate lumbar hernia.1Although abdominal wall hernias have common incidence of 4%-5% globally, lumbar hernias are rare.4 Superior triangle is delineated by 12th rib superiorly, quadratus lumborum medially, internal oblique laterally, floor and roof by external oblique and transversalis fascia.5 There is no shielding action of external oblique on transversalis fascia and presence of subcostal neurovascular bundle results in weakness of the triangle.Lesshaft described superior lumbar hernia in 1870 and Grynfeltt in 1886.We report a case of a 66-years old male who presented with a swelling in the left lumbar region for ten years. The swelling was insidious in onset and was gradually increasing in size. The patient also complained of vague dragging pain for the last two years. The patient denied any history of trauma or any other comorbidities.th rib. It was globular in shape, non-tender, non-mobile, irreducible with a well-defined border and smooth surface. The overlying skin was normal, and a visible cough impulse was present. The swelling was not ballotable and was non-pulsatile.On examination, his general condition and vital signs were unremarkable. On local examination, a 10x13cm swelling  was presRoutine laboratory tests were unremarkable. Contrast-Enhanced Computed Tomography (CECT) of Abdomen and Pelvis showed a 4cm defect in the left lumbar region, with herniation of perinephric fat (10x5cm) with no bowel loops suggestive of superior lumbar hernia .Laparoscopic transperitoneal mesh repair was planned. The patient was kept supine, and pneumoperitoneum was created using Hassan's open technique via a 10mm umbilical port. Two additional ports were created under direct vision in the left hypochondrium and the left iliac region .The defect was visualized by dissecting omental adhesions and entering the retroperitoneal space by incising the peritoneum over the defect. The location of the defect was also guided by external palpation of the hernia. A left superior lumbar hernia with herniation of perinephric fat was noted intraoperatively .Prolene mesh of 15x15cm dimension was placed over the defect and fixed with absorbable tackers, followed by the closure of the port sites with polyglactin 910 sutures. The postoperative period was uneventful, and the patient was discharged on the following day. On one week of follow-up, the patient was asymptomatic without any dragging pain in the lumbar region.3 Increase in intra-abdominal pressure is the most common cause of primarily acquired lumbar hernia, whereas trauma, prior surgical incisions, and abscess formation cause secondarily acquired hernia.Primary lumbar hernia is a rare entity, and the diagnosis in the above case was facilitated by a high degree of clinical suspicion and was confirmed by computed tomography. A lumbar hernia is a protrusion of intraperitoneal or extraperitoneal contents through a defect of the posterolateral abdominal wall.6 Lumbar hernias are commonly unilateral, occurring on the left side mainly between fifth and seventh decades with a Male:Female ratio of 2:1.4Lumbar hernias can be congenital (20%) or acquired (80%). An acquired hernia can either be primary or secondary. The cause of secondary lumbar hernias can be traumatic or post-surgical following trauma, incision, inflammation comprising about 25% of acquired hernia. Congenital hernias arise from the inferior triangle and can be associated with other anomalies like renal agenesis, lumbo-costovertebral syndrome.7 Patients are usually asymptomatic but can present with dull aching pain over the flank region. At present, there are only few literatures regarding pain management of lumbar hernia though operative being the main line treatment. Somatic pain may present with localized or referred pain to either anterior abdominal wall or through sciatic nerve. The main line management of this pain would be non-opioids agents like non-steroidal anti-inflammatory drugs and acetaminophen. If pain still persists, even after non-opioids treatment weak opioids need to be started.8Early detection and management are of paramount importance as up to one out of four cases will become incarcerated, and about one out of ten cases may get strangulated over time.1 Intestinal obstruction, incarceration, strangulation, and volvulus may complicate lumbar hernias.6 Clinical diagnosis maybe difficult, especially in patients who are obese or have non-specific symptoms.Mass can be prominent on straining and coughing and reduced on prone position. The patient might also complain of back pain. There can be symptoms of bowel obstruction manifesting with nausea, vomiting, and abdominal distension. If there are renal contents patient presents with urinary complications such as hematuria, oliguria, and colicky pain.th rib which was non-tender, non-mobile, irreducible with visible cough impulse. So, lumbar hernia was one of the differential diagnoses in this case. Lumbar hernias are frequently misdiagnosed as lipomas; however, they can also be mistaken for a lumbar hematoma, sarcoma, fibroma, abscess, and renal masses. A Contrast-Enhanced Computed Tomography (CECT) scan is the gold standard for confirming the diagnosis of lumbar hernia.9 The diagnosis in our case was also confirmed by CECT of Abdomen and Pelvis when it showed a 4cm defect in left lumbar region with herniation of perinephric fat.As in our case, the patient presented with left lumbar swelling for the last 10 years with no significant history of trauma or surgeries. On local examination, 10x13cm swelling was present below the 126 Only 9% of lumbar hernias present as surgical emergencies while 91% cases are nonemergency cases. Burick AJ, et al. introduced the first trans-abdominal laparoscopic approach.12 In Woodward, et al. used balloon dissector for the totally extraperitoneal approach.13 A prospective study in between January 1997 and January 2003 of lumbar hernia repairs comparing different approaches, the laparoscopic approach showed statistically significant lower morbidity rates, short hospital stay, reduced analgesics requirement, and early return to everyday activities.12 Synthetic mesh for hernia repair may have complications of infection, fistula, and bowel obstruction.Although lumbar hernia is a rare entity, the surgical procedure for lumbar hernia has constantly been evolving since it was first reported by Owen in 1888.1 Even though various operative techniques are described, there is no consensus on the best approach.1 Open hernia repair, which has been used as a conventional approach, is proven safe and effective. With the development of laparoscopic technology, laparoscopic herniorrhaphy is increasingly used to treat a lumbar hernia.13 Operative management needs to be decided based on the size of hernia, site, contents, and availability of facilities and expertise of the hospital. For failed laparoscopic approaches and large defects, open surgery may be recommended.1 To avoid post operative pain, nerves around superior lumbar triangle such as the ilioinguinal nerve, lateral femoral cutaneous nerve and genitofemoral nerve, needs to be protected, during the operations, especially during fixation.14Laparoscopic transperitoneal mesh repair was done in our case. The postoperative hospital stay was uneventful. Follow up was done after a week and the patient was asymptomatic without any dragging pain in the lumbar region. Synthetic mesh for hernia repair may have complications of infection, fistula, and bowel obstruction. Biosynthetic mesh made of the human acellular dermis has been used in some centers, with comparatively good results, especially in contaminated wounds.Primary lumbar hernia is a rare entity and is often misdiagnosed as another lumbar swelling like lipoma, cold TB abscess, and pseudo hernia due to muscular paralysis. A lumbar hernia needs to be considered if the patient presents with pain and swelling over the lumbar region. A Contrast-Enhanced Computed Tomography scan is the gold standard for diagnosing the lumbar hernia. All the lumbar hernia needs to be repaired either by laparoscopic mesh repair or open surgery.


Uterocutaneous fistula is a rare complication that occurs after cesarean section and other pelvic operations. Here we report a case of a 27 years woman presented to our department with a mass and pus-like discharge coming from her previous Pfannenstiel incision for 1 month. The definitive treatment of such cases is hysterectomy but the case was managed by fistulectomy along with gonadotropin-releasing hormone agonist. We report a very rare case of post-cesarean uterocutaneous fistula that was successfully treated with excision of the fistulous tract from the skin and repair of the fistulous uterus.Uterocutaneous fistula is a rare condition where communication is between uterus and skin.th post-op day, the patient developed a Surgical site infection (SSI) and managed with daily dressing, oral antibiotics, and resuturing done after 2 weeks. She commenced on gonadotropin-releasing hormone agonist (GnRH) analog i.e. injection Leuprolide acetate 3.75mg intramuscular monthly for 6 months. Follow-up USG done after 1 month, showed no fistula tract and she was kept on monthly visits till 6 months where she had no complaints.A 27 years old woman presented to our department with mass and pus-like discharge coming from her previous Pfannenstiel incision for 1 month. She underwent lower segment cesarean section (LSCS) at a different hospital for fetal distress 6 months back. She was treated conservatively but she didn't improve. Ultrasound (USG) and Magnetic resonance imaging (MRI) were done which reported an 11-12mm fistulous tract extending through the muscular plane of the uterus. She was scheduled for a laparotomy but a patient came to our center for a second opinion. After counseling, the patient was planned for laparotomy with excision of the fistula tract and/or hysterectomy if required. Surgery was performed with a team of gynecologists and general surgeons. Intraoperatively, Methylene Blue dye was injected from the fistula opening and the tract was followed. Excision of the fistula tract was done and an 8f foley catheter was kept in the uterine cavity for 2 weeks. The immediate post-operative conditions were satisfactory. On the 103 It is a rare condition where the pathophysiology has not been clearly defined but documented following lower segment cesarean section, Infection, Inflammation, Postpartum sepsis, Invasive endometriosis, and uterine procedures.4 Thakur, et al. reported a 30-year old lady, who developed uterocutaneous fistula after undergoing emergency lower segment cesarean section for failed induction 2 months back and developed a massive primary postpartum hemorrhage at the time of cesarean section which was managed with B-Lynch suture and vessel ligation5 and Lim, et al.6 reported a 33-year-old lady who had an emergency cesarean section for retained second twin which was complicated by utero-cutaneous fistula due to red degeneration of intramural fibroids.Uterocutaneous fistula is an abnormal communication between the epithelial lining of the skin and uterus.7 reported uterocutaneous fistula following lower segment cesarean section.Jain, et al.8 reported uterocutaneous fistula which developed following septic abortion induced by laminaria tent insertion in the cervix. Because of the rarity of uterocutaneous fistula, there is no available evidence-based treatment modality. In the literature, the delay between last surgical event and occurrence of fistula ranges between 2 months to 6 years. The diagnosis of uterine fistula can be straightforward with pathognomonic clinical signs such as bloody discharge through abdominal scar during menstruation. Other investigations include - Fistulography with Injection of contrast material through skin opening showing connection to the uterus.1Gupta, et al.1 A standard treatment has yet to be introduced. Recent studies suggest combining surgical and medical treatment to avoid the risk of hysterectomy.Hysterosalpingography and MRI with contrast is another modality.9 However in another case report of uterocutaneous fistula following term abdominal pregnancy, promsonthi and Herbutya performed a subtotal hysterectomy.10 Seyhan, et al.11 reported a patient treated with GnRH agonist alone which induces atrophic changes in epithelium and assist in the closure of the fistula. Thubert, et al.1 used medical treatment and minimally invasive surgery for excision of the fistula tract. Although there is no evidence-based treatment modality currently available for utero-cutaneous fistula, various treatment options have been reported in the management of utero-cutaneous fistula where surgery has remained the treatment of choice. A combination of both medical and surgical treatment can be advantageous and averts the risks associated with major surgeries.3In a case report of endometriotic uterocutaneous fistula following cesarean section, Dragoumis, et al. carried out a total abdominal hysterectomy in a 44 years old lady.This case report highlights the very rare possibility of uterocutaneous fistula occurring in a woman following cesarean section one month back. Appropriate surgical skills and postoperative care are necessary to prevent an outcome that may be agonizing for the patient. While there is no evidence-based modality of therapy, further research is necessary to explicate the mechanisms of this fistula following cesarean section. Imaging studies are instrumental in the evaluation of cases of uterocutaneous fistula.


Solitary lipomas are the most common soft tissue tumor. Lipomas include a gamut of subtypes. Spindle cell lipoma (SCL) is a distinct entity accounting for about 1.5% of all lipomasA 48 year old male reported with a history of a left sided neck swelling since 2 years which was progressively increasing in size. A 4x4 cm swelling was present in the anterior triangle of the neck which was firm and mobile. ENT examination and investigations were unremarkable.Fine needle aspiration cytology showed loose arrangement of spindle cells in the myxoid matrix, suggestive of a soft tissue lesion. On computed tomography scan a 3.2x4.2 cm sized soft tissue density was seen in close proximity to the superficial lobe of the left parotid with tiny hypodensities of fat attenuation in the periphery. The lesion was extending anterior to the sternocleidomastoid muscle displacing the platysma. Post contrast showed a heterogenous enhancement. Figure 1cPatient underwent surgical excision of the mass. The mass was subcutaneous well defined and encapsulated. The mass was excised completely.On histopathological examination the tumor was composed predominantly of spindle cells which were uniform with an elongated nucleus with inconspicuous nucleloli. The cells were arranged in short parallel bundles. The background showed mucoid matrix mixed with ropey collagen fibre bundles. Mature adipocytes were also seen. On immunohistochemistry CD 34 antigen was positive and a diagnosis of spindle cell lipoma was made.One year post operatively there is no evidence of a recurrence.Enzinger and Harvey first reported on SCL in 1975The radiological appearance is not pathognomic. The variation in the ratio of fat and spindle cell causes the wide spectrum of imaging features. On T1 weighted MRI the lesion is isointense to the subcutaneous fat. T2 weighted MRI with fat suppression reveals a hypointense lipomatous component and a hyperintense spindle cell component which shows enhancement on contrastHistologically SCL consists of a mixture of bland spindle cell and mature adipocytes. The matrix is composed of varying amounts of mucoid material and collagen. The spindle cells have scant cytoplasm, elongated nuclei and are arranged in short parallel arrays. The amount of adult fat is variable and lipoblasts are generally not seenPleomorphic lipoma which is considered a highly pleomorphic variant was first described by Shmookler and Enzinger, as both of these have similar cytogenetics they are considered as a single entityIt is important to differentiate SCL from liposarcoma. Liposarcoma are characterized by more numerous spindle cells, presence of nuclear pleomorphism and MDM-2 amplificationThe treatment is surgical excision and recurrences are extremely rare even with incomplete excision.Spindle cell lipoma is an extremely rare variant of the common lipoma. The condition is benign and careful identification of the lesion is necessary to avoid misdiagnosing it as its malignant counterpart. Treatment involves surgical excision with virtually no recurrences.


Chronic spinal epidural hematomas (SEHs) are rare clinical entities. SEH with vertebral scalloping is extremely rare, with only a few cases having been reported to date. We report a unique case of spontaneous chronic SEH in the lumbar spine with severe vertebral scalloping mimicking an epidural tumor.A 71-year-old man presented with a 2-month history of lumbar pain and a 3-week history of paresthesia and pain in the right lower extremity, hypesthesia in the perineal and perianal regions, and bladder dysfunction. Computed tomography following myelography revealed an epidural mass lesion on the right side that compressed the dural sac and was associated with severe bony scalloping on the posterior wall of the L4 vertebral body. Magnetic resonance imaging (MRI) on T1- and T2-weighted images revealed a space-occupying lesion with heterogeneous intensity, and T1-gadolinium images showed an intralesional heterogeneous enhancement effect. A tumoral lesion in the spinal canal was suspected, based on preoperative imaging; therefore, a total spinal tumor resection was planned. Intraoperative findings revealed that the brownish lesion adhered to the dura and epidural tissues in the spinal canal, and the space-occupying mass in the scalloped cavity of the posterior wall of the L4 vertebra was encapsulated in red-brownish soft tissues. The lesion was totally resected in a piecemeal fashion, and pathological examination revealed a mixture of tissues that contained a relatively new hematoma with hemoglobin, as well as an obsolete hematoma with hemosiderin and amyloid deposits. The mass was diagnosed as a chronic epidural hematoma with recurrent hemorrhage. The postoperative course was uneventful, and the preoperative neurological symptoms immediately improved.The preoperative diagnosis of chronic SEHs is challenging, as MRI results may not be conclusive, particularly in patients with scalloping of bony structures. Thus, chronic SEHs should be considered as a differential diagnosis in cases of suspected tumoral lesions in the spinal canal. To the best of our knowledge, this is the first reported case of acute exacerbation of chronic SEH with cauda equina syndrome and severe vertebral scalloping. Spinal epidural hematomas (SEHs) are uncommon clinical entities, accounting for only 0.3–0.9% of all space-occupying lesions in the spinal canal , 2. ChroA 71-year-old man presented to our hospital with a 2-month history of lumbar pain and a 3-week history of paresthesia with pain in the right lower extremity, hypesthesia in the perineal and perianal regions, bladder dysfunction , and intermittent claudication. He had a history of cerebral infarction 8 years prior and was treated with antiplatelet therapy (clopidogrel). All blood and coagulation tests  revealed normal values. There was no history of major or minor spinal trauma. On physical examination, the patella tendon reflex was normal bilaterally. The right Achilles tendon reflex was diminished, and the left Achilles tendon reflex was normal. Babinski’s sign was absent bilaterally. He had no motor weakness in his bilateral extremities.MRI performed before hospitalization revealed a space-occupying lesion that extended from the right intravertebral region to the intraspinal canal at the level of the L4 vertebra on the right side. The mass lesion in the spinal canal had heterogeneous intensity on T1-weighted images and low intensity on T2-weighted images. Gadolinium-enhanced T1-weighted images showed a heterogeneous enhancement effect in the lesion  improved immediately after the operation. He was discharged on the 11th postoperative day and was able to walk without aid. The patient was able to resume his previous job delivering newspapers at 2 months postoperatively. Good clinical outcomes were maintained at the final 2-year postoperative follow-up, with MRI confirming the disappearance of the epidural space-occupying lesion Fig. a, b.Fig.SEH can be classified as idiopathic, spontaneous, or secondary, based on its pathogenesis , 2. IdioThe pathophysiology of chronic idiopathic or spontaneous SEH is not clear. One hypothesis is the rupture of Batson’s plexus due to variations in abdominal or thoracic pressure –7. BatsoPrevious studies have reported that chronic SEHs often result in a lumbar radiculopathy with a chronic clinical course , 8. To dThe histological examination revealed relatively old hemorrhagic changes (hemosiderin deposition), in addition to an early phase change (granulation tissue and blood clot). This implied that a serial bleeding process was responsible for the gradual formation and expansion of the space-occupying lesion and subsequent scalloping. The cauda equina symptoms may have been induced by the recurrence of this bleeding process.MRI may be considered to be the diagnostic standard for SEHs at a certain level; however, MRI signal intensities of SEHs are variable and depend on the age and stage of biochemical evolution of the hematoma , 10, 11.It is well known that the increased intraspinal pressure exerted by a slowly expanding intraspinal tumorous lesion may cause scalloping of the spinal osseous structures. Schwannomas, dermoid cysts, epidermoid cysts, and lipomas occasionally demonstrate vertebral scalloping –18. In aIn conclusion, chronic SEHs are rare clinical entities that can be challenging to diagnosis via preoperative imaging, particularly when associated with vertebral scalloping. Although rare, SEHs should always be considered as a differential diagnosis in patients with suspected tumoral lesions in the spinal canal. Furthermore, surgeons should be aware that chronic SEHs can cause vertebral bone erosion.


Leydig cell tumor (LCT) is a sex cord-stromal tumor, which is a clinically rare ovarian tumor. It is characterized by endocrine hormonal changes and usually occurs in postmenopausal women.We report the clinical case of a 38-year-old female of childbearing age with LCT of the right ovary who presented with significantly decreased menstrual flow and elevated androgen levels, with persistent hypoechoic areas in the ovary as demonstrated by transvaginal ultrasound.The transvaginal ultrasound suggested the presence of a hypoechoic area in the right ovary with elevated androgens, interstitial tumor of the ovarian sex cord may be considered.The patient underwent laparoscopic right adnexectomy.Postoperative pathology confirmed the morphology and immunohistochemistry of the right adnexa consistent with LCT, and no areas of malignant transformation were found on multiple sections of the surgical specimen. The patient had normal androgen levels at postoperative day 2, day 45 and month 3. There was no sign of recurrence.This case suggests that when women of childbearing age have abruptly decreased menstrual flow with increased testosterone, clinicians should pay attention to intra-ovarian occupying lesions and consider the possibility of LCT. In such cases, ultrasound examination can determine the presence, location, shape and size of occupying ovarian lesions and play an important role in the diagnosis of condition. However, because of the very rare incidence of LCT and the few published case reports, gynecologists are usually unfamiliar with this type ovarian tumor. In addition, LCT may be asymptomatic and the ovarian masses in these patients are usually small and difficult to detect by gynecologic examination, which makes them difficult to diagnose. Ultrasound is a dynamic, real-time, noninvasive examination technique that is highly sensitive for the diagnosis of ovarian masses and is an effective tool for the evaluation of ovarian masses that can be widely used. We report a 38-year-old female with LCT whose gynecologic ultrasound suggested a persistent hypoechoic area in the right ovary with abundant blood flow around it. Our case is a useful addition to the diagnosis of LCT ultrasound in women of childbearing age.A 38-year-old woman, G1P1, last menstrual period: 2022-05-05, used to have regular menstruation, 5 to 6/30 days, with moderate volume and no dysmenorrhea. Six months ago, there was a change in menstruation without any obvious trigger, the cycle became 10 to 45 days, the period was shortened to 0.5 to 1 day and the volume was very low. This study was approved by the First Hospital of Hebei Medical University, and informed written consent was obtained from the patient for publication of this case report.Transvaginal ultrasound: the endometrial thickness was about 0.35 cm, and a hypoechoic area of approximately 1.49 × 1.20 cm in size was found in the right ovary, with abundant blood flow around it. On postmenstrual review: the endometrial thickness was about 0.37 cm and a hypoechoic area of about 2.03 × 1.52 cm in size with abundant blood flow around it were found in the right ovary ,,8 and usually rich blood flow can be detected by ultrasound. The ultrasound presentation of this disease has been less reported in the previous literature. In this case, the ultrasound image suggested a persistent hypoechoic area in the right ovary with abundant blood flow around it, which was considered an ovarian lesion. This provided an important help for the clinician to determine the patient’s condition.Imaging ancillary examinations are very important in diagnosis, and ultrasonography is a noninvasive, convenient, efficient, and mature examination modality that has become the preferred method for adnexal tumor diagnosis because of its high sensitivity, good acceptability, and low cost. However, MRI is not preferred in clinical practice because of its high cost.Of course, there are some limitations of ultrasonography which cannot identify lesions that are too small and has poor visualization of the subtle structures within the lesion, resulting in such masses can easily be missed or mistaken for a corpus luteum. Therefore, when the patient’s androgens are significantly higher than normal, even a normal ultrasound presentation cannot exclude the diagnosis of LCT. MRI has a high soft tissue resolution and is also meaningful for the detection of ovarian tumors when clinically indicated, but no abnormalities are seen on ultrasound.,14 with the appearance of masculine features as the main symptom. In this case, the patient was a female of childbearing age with the first symptom of significantly decreased menstrual flow. After excluding lesions of uterine, pituitary and hypothalamic origin, elevated testosterone and the persistence of the hypoechoic area in the right ovary suggested by ultrasound were important to clarify the patient’s diagnosis and treat the patient with early surgery, and postoperative pathology and immunohistochemistry confirmed that the case was consistent with LCT.We reviewed previous case reports of LCT and found that LCT occurs mostly in postmenopausal women,In conclusion, this case suggests that when women of childbearing age have abruptly decreased menstrual flow with increased testosterone, clinicians should pay attention to intra-ovarian occupying lesions and consider the possibility of LCT. Ultrasonography can help examine the presence, location, shape and size of occupying ovarian lesions, which can assist clinicians in diagnosing the condition and performing timely surgery to relieve the symptoms of hyperandrogenemia and help patients resume normal menstruation.CT contributed to the analysis and interpretation of data and revising of the manuscript for important intellectual content. XL was responsible for data curation. JL contributed to acquisition of additional clinicopathological data. YH was responsible for the acquisition of data, and drafting of the manuscript. ZF was responsible for final approval of the manuscript to be published. All authors contributed to and approved of the final version of the manuscript.Data curation: Cai Tian, Xiaodong Li.Methodology: Xiaodong Li.Project administration: Jingqiao Liu.Writing – original draft: Yiwei Han.Writing – review & editing: Zijie Fu.


The thymus gland is a lymphoid organ normally located in the superior anterior mediastinum. It can rarely present abnormally in other sites along the thymopharyngeal canal and it might cause difficulties in breathing and/or feeding. We present a case report of an ectopic cervical thymus of a 10-month-old male infant who was presented to the hospital with a swelling on the left side of his neck for nine months. Investigations raised suspicion about four differential diagnoses and a total surgical excision for histopathological confirmation was deemed mandatory. It is of great importance to consider ectopic cervical thymus in the differential diagnosis of pediatric neck masses to avoid unnecessary procedures and prevent possible complications. The thymus is a lymphoid organ, located in the superior anterior mediastinum, and is responsible for the maturation and differentiation of T lymphocytes. In neonates and infants, it is larger, more active, and in a state of continuous growth until it reaches its maximum weight (30-40 g) by the age of two to three years. As children grow into adolescence, the thymus begins to involute until it becomes an atrophic gland, mostly replaced by fatty tissue .Ectopic thymus is a rare, typically benign pediatric condition in which the thymic tissue is located at an unusual site along its migration route, known as the thymopharyngeal canal . It occuPatients with ectopic thymus are usually asymptomatic; however, compressive symptoms may occur in some cases, presenting with feeding difficulties, breathing difficulties, or Horner’s syndrome in rare cases ,5. AlthoIn this article, we draw our attention to the diagnosis and management of this rare condition by reporting a case of an ectopic cervical thymus confirmed postoperatively by histopathological examination in a 10-month-old male infant who presented with left neck swelling.A 10-month-old boy presented to the hospital with a swelling over the left side of the neck for over nine months. The swelling was insidious in onset with no change in size. Based on the history of the presenting illness, he had no history of fever, snoring, or any difficulties in feeding or breathing. Family history and past medical history of the patient were insignificant with no history of radiation exposure. He was a full-term neonate with no peripartum complications. Upon physical examination, the patient was vitally stable with normal height and weight. Examination of the neck revealed a left cervical swelling, adjacent to the left sternocleidomastoid muscle measuring approximately 2-3 cm. The swelling was freely mobile, non-tender, non-pulsatile, and non-compressible, with no skin changes. An ultrasound of the neck was performed illustrating a well-defined soft tissue mass in the left upper aspect of the neck, medial to the sternocleidomastoid muscle, and anterior to the carotid bifurcation. It measured 24x15.5mm, with internal vascularity. Further assessment was indicated; thus, an MRI was carried out. It revealed a well-circumscribed lesion on the left side of the neck measuring 3.5 x 2.3 x 2.2 cm in largest diameters. The lesion was located anterior to the sternocleidomastoid muscle, pushing the left submandibular gland anteriorly. It was superficial to the carotid sheath space, slightly pushing it posterio-laterally (Figure Under general anesthesia, total excision of the mass was performed successfully with no perioperative complications, and the excised lesion was sent for histopathological examination. Upon gross examination, the soft tissue mass was solid in nature, oval-shaped, nodular, well-circumscribed, pink to dusky-blue in color, with increased vascularization. Microscopic examination reported that the excised lesion was in fact a thymic tissue with normal architecture and variably sized lobes and lobules. Some of the lobules appeared to have a thinner cortex and were associated with tingible body macrophages indicating mild involution. Small Hassall’s corpuscles were seen in the medulla with calcification and little necrotic debris. The final diagnosis of this case was confirmed to be a solid ectopic cervical thymus.  The patient was doing well and was discharged on day two post-operatively. On follow-up visits, the child was in a good state of health with no apparent complications, recurrence, or any complaints. The thymus is a paired organ that develops from the ventral saccules of the third and occasionally, the fourth pharyngeal pouches during the sixth week of embryonic life. The primordial cells start to move medially and caudally to the thyroid in the eighth week of fetal life. The bilateral thymic primordia descend behind the sternum into the superior mediastinum after they fuse in the midline . It gainClinically, ECT in children presents as a firm, asymptomatic neck mass with no specific clinical features. Only 6% of patients could have symptoms due to compression of the trachea which may result in stridor and dyspnea and/or compression of the esophagus which may lead to dysphagia ,21. It'sUltrasound is considered the first-line imaging modality in diagnosing cervical ectopic thymus, as ECT is characterized by the presence of echogenic flecks in a hypoechoic background known as the “Starry Sky Appearance” . ComputeThe sole confirmatory method for the diagnosis of ectopic cervical thymus is via histopathological examination . The preEctopic cervical thymus is a rarely diagnosed condition that should be considered in the differential diagnosis of pediatric unilateral neck masses to prevent unnecessary diagnostic and therapeutic procedures. Ultrasonography should be the first modality of diagnosis followed by MRI. In difficult cases, fine needle aspiration cytology can be considered to confirm the diagnosis. Although it is rarely symptomatic, surgical excision is recommended to avoid compression symptoms which can lead to fatal outcomes, such as dyspnea and dysphagia. However, the presence of a normal mediastinal thymus gland should be confirmed prior to the surgery to avoid total thymectomy.


Primary malignancies of the nasal cavity and paranasal sinuses account for 0.3% of all head and neck cancers with a proportion of tumor in sphenoid and frontal sinuses estimated to 0.001%.A 67 year-old man was referred to the Department of Otolaryngology for the evaluation of a few days history of monocular diplopia, left facial and frontal pain. Patient did not report fever or continuous rhinorrheas. The general medical history revealed that the patient had previously been treated for a prostatic carcinoma by surgery, radiotherapy and hormonal therapy. The ENT clinical examination has just reported the presence of a divergent strabismus of the left eye and frontal pain increased on palpation. Concerning the nasofibroscopic examination, we did not found rhinorrhea, a nasal extension of the tumor and signs suggesting a complication of a rhinosinusitis. The frontal mass seemed not be complicated by a rhinosinusitis. A first physician suspected the diagnosis of a rhinosinusitis with a possible sinus cavernous thrombosis. Therefore, he prescribed a Magnetic Resonance Imaging (MRI) and a Computed Tomography (CT) that showed two focal lesions, the first in the left cavernous sinus and the second in the left frontal sinus . The lesa ≥ 2 ≥ mm dural thickening, the loss of hypointense zone  and nodular dural enhancement could be highly predictive for a dural invasion6The mechanism of prostatic metastases to the paranasal sinuses involves a spread cells that probably comes from the vertebral venous plexus.Concomitant metastases of the frontal and cavernous sinuses are a rare condition that can lead to a misdiagnosis of rhinosinusitis. In the context of a history of cancer, even if it remains exceptional, an imaging must be made to precise the etiology of the symptoms. In the case of frontal malignancy, dural thickening and nodular enhancement combined with the loss of hypointense zone could be highly predictive for a dural invasion implying a poor prognosis.The authors declare no conflicts of interest.


Paragangliomas (PGLs) are uncommon tumors of uncertain malignant potential. Multifocal paragangliomas are scarcely reported in the literature.A 25-year-old male patient was reported for the first time with multifocal para-aortic and para-vesical PGLs. The diagnosis was identified by blood catecholamine tests and enhanced CT scan and MIBG scintigraphy. A resection surgery was performed for treatment and the immunochemistry test of the tumors presented the features of PGL.A case of multifocal para-aortic and para-vesical PGLs confirmed by the catecholamine test, enhanced CT, and MIBG scintigraphy is presented. The cooperation of experienced surgeons, anesthesiologists, and endocrinologists was critical in treatment. Paragangliomas (PGLs) are uncommon tumors consisting of neural crest cells that originate from the paravertebral sympathetic chains and parasympathetic ganglions of the chest, abdomen, and pelvic cavity . The incMost PGLs secrete catecholamines and present with episodes of high blood pressure, heart palpitations, sweating, and headache. Some may be hormonally inactive, but this is the minority. In such cases, presentation will be due to pain and functional loss due to invasion into neighboring structures and organs. However, much more commonly, many of these tumors are diagnosed incidentally because of medical imaging for other reasons . The metHere, we present a case report of a patient with multifocal para-aortic para-vesical PGLs.A 25-year-old male was admitted to the Urology Ward of West China Hospital, Sichuan University, in March 2022. One month ago, a tumor close to the urinary bladder was incidentally found during ultrasonography in routine physical examination. The records at admission showed that the blood pressure of the patient was 126/73 mmHg, and the heart rate was 80 bpm when he was inpatient. No other chief complaints or marked symptoms were presented, such as headaches, blood pressure turbulence, or abdominal pain. The patient had no history of hepatitis and tuberculosis, nor did he have a history of allergies, trauma, surgery, familial disease, alcoholism, or smoking. The physical examination showed a soft abdomen, and no mass, regional lymphadenopathy, or pain was reported.131I-mataiodobenzyl-guanidine (MIBG) scintigraphy results showed two radio-uptake enhancing regions in the left abdomen and pelvis after 48 and 72 h . The tumor adjacent to the bladder measured 4.0 × 3.1 × 2.5 cm, and the para-aortic tumor measured 5.6 × 5.2 × 4.6 cm. The patient’s postoperative course was uneventful and the patient was discharged 4 days post-surgery.The final diagnosis was confirmed by histopathology of the specimens: The envelopes of the tumors were intact; the cystic-solid tumor close to the aorta presented a grey-white nodular morphology with hemorrhage and necrosis, cysts were individual and smooth, with yellow-brown tissues inside   . The MIBThe pathology mechanism of PGLs varies. One such mechanism is that the Nfr2 activation and the elevated glucose uptake contribute to PGL malignancy with SDHB gene mutation . VHL and131I-MIBG therapy is well adopted in PGL patients and reduces the catecholamine levels  for the publication of any potentially identifiable images or data included in this article.Conceptualization: XL. Data curation and Project administration: S-CX and KW. Staining slides provision: M-NZ. Manuscript Writing—Original draft: S-CX and X-PD. Manuscript editing and manuscript review: XL and X-PD. This manuscript has been read and approved by all the authors.This work was supported by the Sichuan Science and Technology Program (2022YFS0133).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.


Primary retroperitoneal mucinous cystic tumours with borderline malignancy (PRMC-BM) are rare and difficult to diagnose preoperatively. We are the first to report two cases of PRMC-BM which mimic a duplex kidney and evaluate the outcomes of different surgical procedures.We describe two cases of retroperitoneal cystic tumours. Both were diagnosed with duplex kidney with hydronephrosis on computed tomography scan. The first patient underwent robot-assisted laparoscopic surgery and was found to have a retroperitoneal cystic tumour. The other patient underwent an ultrasound-guided puncture before surgery and was diagnosed with retroperitoneal lymphangioma. Retroperitoneal cystectomy was performed using an open transperitoneal procedure. The final pathologic diagnosis in both cases implies PRMC-BM. The open surgical approach was associated with a shorter operation time, less intraoperative blood loss, and protected cyst wall integrity by comparing the different surgical approaches. During follow-up, the patient in the first case had tumour recurrence six months post-surgery, and the other patient was healthy without recurrence or metastasis 12 months post-surgery.Primary retroperitoneal mucinous cystic tumours with borderline malignancy can be enclosed within the kidney and misdiagnosed as other cystic diseases of the urinary system. Thus, an open surgical approach may be more suitable for this type of tumour. Primary retroperitoneal mucinous cystic tumour with borderline malignancy (PRMC-BM) is an extremely rare disease . Only 23A 56-year-old woman presented with painful urination and discomfort in the right waist for one year. Examination revealed irregular cystic masses  in the right abdomen. Abdominal computed tomography (CT) revealed irregular low-density lesions measuring approximately 28 × 22 × 8 cm in the right ureter area, beginning from the lower pole of the right kidney to the bladder neck. Contrast-enhanced CT showed obvious enhancement of the cyst wall, but the borderline of the lesion and the lower part of the right ureter were unclear during the renal excretion stage Fig. a. The prA partial nephrectomy for a duplex kidney was scheduled using a robot-assisted laparoscopic approach. The trocars were successfully established. After the lateral peritoneum was opened, we found an independent cystic mass close to the right kidney, rather than a duplicated kidney malformation. The tumour cyst wall was then opened and a cream-like liquid was observed. Another cystic mass was found under the tumour. Both lesions were removed.The operation time was 470 min, and intraoperative blood loss was approximately 600 mL. The patient recovered well and was discharged four days postoperatively without any postoperative complications. The final pathological diagnosis was PRMC-BM Fig. c–f. DuriA 68-year-old woman was admitted to our hospital with a mass in the lower left retroperitoneum. A physical examination revealed a local bulge in the left lower abdomen, and a palpable mass approximately 15 × 10 cm in size. The mass was tough and could be moved without tenderness. Abdominal contrast-enhanced CT revealed a cystic mass with the size of 15 × 11 × 9 cm below the left kidney. The borderline between the cystic mass and the left ureter was unclear. The left kidney showed a double renal pelvis and ureter accompanied by hydronephrosis and dilatation of the ureter. There was no obvious enhancement of the cyst wall or cyst Fig. a. It wasRetroperitoneal mass resection was performed using an open approach. Before tumour resection, a double J was placed in the left ureter. A transverse incision of approximately 15 cm in length was made on the left abdomen. After exploring the peritoneum, a large cystic mass was observed below the lower pole of the kidney. The mass surrounded the ureter and was underneath the renal hilum. The mass was completely removed without secondary injury to the cystic wall. The duplicated ureters were intact Fig. b. The cyThe operation time was 210 min, and intraoperative blood loss was approximately 30 mL. The patient recovered well and was discharged six days postoperatively without any complications. The final diagnosis was PRMC-BM (Fig. Mucinous cystadenomas are common in the ovaries, pancreas, and appendices. It is thought to be a PRMC when located in the retroperitoneum with a normal visceral organ. AccordiPreoperative diagnosis of PRMC is very difficult. Imaging is helpful in the detection of retroperitoneal cystic masses, but it is difficult to differentiate PRMC from a variety of retroperitoneal cystic diseases because of the lack of typical imaging features. In the The occurrence of PRMC is most commonly seen in female patients, but its histogenesis remains unclear. The main theories are as follows: seeding of ectopic ovarian tissue, monoderFurthermore, PRMC-BM is a potential malignant type of PRMC. However, we reviewed other reports and found that the recurrence rate in such cases was very small. We tried to analyse the imaging features of the cystic lesions, multilocular, enhanced, and solid nodules but failed to reveal any regularity in the recurrence rate. Chemotherapy is recommended for mucinous cystadenocarcinoma. HoweverIn conclusion, PRMC-BM is a rare disease that may mimic urinary tract-related diseases, and urologists need to be aware of it. Ultrasound-guided puncture of the cyst may be useful in differential diagnosis before surgery. Since cystic masses can be malignant, careful protection of the cyst wall and prevention of cyst rupture can reduce the risk of tumour recurrence, and an open surgical approach may be more appropriate.


Carcinoma ex‐pleomorphic adenoma (Ca ex‐PA) comprises 0.5% of head and neck neoplasms. Transoral robotic surgery (TORS) is an approach being used to treat a variety of benign and malignant head and neck neoplasms. Recently, this technique has gained popularity as an alternative for parapharyngeal space (PPS) tumor resection. To our knowledge, this is the first case of Ca ex‐PA managed successfully by TORS of the PPS.Fifty‐nine‐year‐old male with incidental mass in PPS, initial diagnosis of pleomorphic adenoma, who underwent transoral robotic resection. The histopathology diagnosis with minimally invasive Ca ex‐PA findings and malignant component of high‐grade epithelial/myoepithelial carcinoma and salivary duct carcinoma. Patient discharged on the fifth post‐operative day without complications.Based on our findings, TORS may be a safe procedure to remove selected Ca ex‐PA from the PPS; however, further research is needed. The tumor was resected completely by TORS. The outcome reflected that this technique allows for easily achieved 3D visualization of the tumor in the PPS, shorter operative times, increase in range of motion in the narrow operative field, and a complete pathological specimen removal. Our literature review suggests that this is the first case of Ca ex‐PA of PPS successfully managed by the TORS.2A 59‐year‐old man arrived at a rural hospital in Colombia in November, 2020 after sustaining a scorpion bite on the left foot and was treated accordantly. The patient developed edema and swelling on the left side of the neck; he was treated for cervical suppurative adenitis with surgery and antibiotics.During the post op follow up 1 month later, a CT scan of the neck was obtained reflecting incidental findings of a mass in the right PPS followed by a CT‐guided biopsy. The pathology report indicated epithelial/myoepithelial neoplasia compatible with pleomorphic adenoma Figure . With thA new neck CT scan was taken upon arrival revealing a 40 × 29 mm rounded mass located in the prestyloid space Figure . FlexiblAfter a thorough review of the biopsy by an oncologic pathologist, a medical board meeting was held at which it was concluded to be a benign pleomorphic adenoma; it was decided to perform robot‐assisted transoral resection in July 2021.2.12.1.1Under general anesthesia and after nasotracheal intubation, the da Vinci robotic®  system was docked above the patient's head. Then, a Crowe‐Davis mouth gag, 8 mm HD telescopic camera 0° and 5 mm EndoWrist® instruments were placed. The surgical assistant located at the head of the table performed suction, retraction, and the operating physician sat at the surgeon's console. After conventional right tonsillectomy, a transfixion medial incision was made between the anterior and posterior tonsil pillars, being the superior constrictor muscle the upper edge; the PPS was acceded, and the tumor was identified Figure . A BrainSurgical pathology demonstrated a minimally invasive carcinoma, 2.8 × 1.7 × 0.4 cm ex‐pleomorphic adenoma with a malignant component of high‐grade epithelial/myoepithelial carcinoma and salivary duct carcinoma. Microscopic examination showed the presence of necrosis and mitotic activity of at least 7 mitoses in 10 high‐power fields. No lymphovascular invasion or perineural invasion was observed; the resection edges were negative, with neoplastic infiltration of the lesion capsule with 1 linear mm of tumor invasion Figure . ImmunohDue to the high‐risk tumor reported, he underwent adjuvant IMRT technique external radiotherapy with 1.8 Gy to 54 Gy per fraction in low‐risk volume  and simultaneous integrated reinforcement in a 2.2 to 66 Gy per fraction in high‐risk volume (tumor bed) ending in November 2021. A follow up image was taken 8 weeks later, without tumor persistence Figure . The pat3The surgical treatment method for this patient was TORS technique, considering the multiple studies that have reported its advantages in the treatment of benign and malignant tumors of the oropharynx and larynx,Currently, in the literature there are 32 PPS Ca ex‐PA reported cases,The surgical approach depends on tumor localization, size, initial biopsy, and compromise of adjacent structures by the neoplasia. Patients with PPS tumors must meet the following criteria to be considered as candidates for surgical removal with TORS approach at our institution: have well‐circumscribed tumors up to 8 cm in size,The importance of studying radiological images before surgery guides not only the question of resectability but the surgical approach to be used.When a patient is suitable for transoral approach, the preferred method at our institution is TORS over transoral endoscopic surgery, as it not only allows greater movement in such a narrow working space, but also provides more precision in achieving resection margins due to its wide near‐field visualization.There is lack of information regarding oncological results in this type of tumors, given the rarity of Ca ex‐PA, and in the vast majority of cases its identification is in the surgical specimen, after surgeries without oncologic intention and very conservative.From what has been described in retrospective series in parotid tumors, resections with extracapsular dissection in Ca ex‐PA with minimal capsular infiltration or low‐grade tumor seem to be sufficient without affecting survival or postoperative morbidity.Traditional open methods, in contrast to minimally invasive approaches such as TORS, have the disadvantage of requiring external incisions and extensive dissection of unaffected anatomical structures.Recently, transoral approaches have undergone a complete renovation with robotic surgery since it has great advantages such as a wide near‐field visualization, providing excellent image quality.However, there are some key considerations to keep in mind for the TORS approach to be contraindicated: if the tumor appears infiltrative radiographically, has a poorly defined plane with respect to the great vessels, or displaces the internal carotid medially; in these cases, a transcervical approach should be considered.According to the guideline recommendations on the adjuvance in malignancy of the salivary glands,4Based on the results obtained in this case, we have achieved an effective approach for the resection of a PPS located Ca ex‐PA case without complications. The wide near‐field 3D visualization, shorter operative times and, greater range of motion in narrow spaces are some of the advantages of this technique that contributed to its success while also minimizing bleeding, and avoiding morbid open surgery; as far as limitations we can point out a piece of equipment that is not always available (the da Vinci robotic®) and a prolonged surgical exposure to its use by surgeons to improve results. To our knowledge, this is the first documented case of minimally invasive surgery in Ca ex‐PA by TORS; this is a promising technique, though further studies are still needed.Enrique Cadena‐Piñeros: Conceptualization ; data curation ; formal analysis ; investigation ; methodology ; resources ; supervision ; validation ; visualization ; writing – original draft ; writing – review and editing . Andrey Moreno‐Torres: Data curation ; validation ; visualization ; writing – review and editing . Jessica Correa‐Marin: Conceptualization ; data curation ; formal analysis ; investigation ; methodology ; project administration ; resources ; software ; supervision ; validation ; visualization ; writing – original draft ; writing – review and editing . Mario Alexander Melo‐Uribe: Validation ; writing – original draft ; writing – review and editing .This research did not receive any specific grants from funding agencies in the public, commercial, or not‐for‐profit sectors.The authors have stated explicitly that there are no conflicts of interest in connection with this article.Authors confirm that all procedures followed, were in accordance with the ethical standards with the Helsinki declaration of 1975, as revised in 2000.Informed consent was obtained from the patient for the publication of the details of his case and the use of clinical and personal images.


Curvularia species of fungi are ubiquitous and mostly comprise plant or soil residents. Rarely pathogens, they are found in tropical and subtropical climates. On rare occasions, these fungi can be of clinical significance and lead to a variety of disease processes, mainly in immunocompromised individuals. Most infections are limited to allergic fungal rhinosinusitis; however, disseminated disease and invasive infections have been increasingly reported. There exist no therapeutic guidelines for invasive Curvularia infections currently, but amphotericin and various azoles have been used with varying degrees of success. We present a unique case of an immunocompetent 44-year-old female who presented with symptoms concerning for pneumonia and was found to have a mass-like lesion in the lung concerning for malignancy. Biopsy and histopathology of the lesion were consistent with invasive Curvularia pulmonary infection. We reviewed this case in the setting of reported literature concerning Curvularia with an emphasis on the epidemiology, pathology, diagnosis, and emerging management protocols of invasive Curvularia infections. Curvularia fungal infections are rarely seen in humans. There are currently over 100 identifiable species within the Curvularia genus, but only a few have been reported to cause diseases. The primary species associated with human infections include C. aeria, C. lunata, and C. geniculata. Curvularia species are classified into a group of pigmented fungi called dematiaceous fungi and are characterized by their brown/black pigment and rapid growth in colonies [Curvularia species have low inherent virulence; however, fungal pulmonary infections are increasing in a growing immunosuppressed population. Pulmonary infections with these fungal species are very rare in immunocompetent individuals and specific risk factors in this population have yet to be identified. Clinical manifestations can vary from asymptomatic pulmonary nodules to PNA and endobronchial lesions causing hemoptysis [moptysis . There amoptysis .This article was previously presented as a case report poster at the 2020 CHEST Annual Meeting held from October 18 to 21, 2020.A 44-year-old female with a past medical history of 15 pack-years cigarette use but currently with eight years of abstinence presented to her primary care center with shortness of breath, cough, and purulent sputum for over three to four weeks. The patient was initially diagnosed with pneumonia and treated with levofloxacin, but her symptoms worsened. She presented to an outside hospital and a CT scan of her chest revealed a 5.8-cm right middle lobe mass-like lesion concerning for malignancy Figure . The patCurvularia species. She underwent treatment initially with fluconazole for two weeks. Once susceptibilities returned from the reference laboratory, the patient was switched to posaconazole for a total of six weeks of therapy. After the completion of therapy, the patient had resolution of her symptoms, and a repeat CT scan of her chest revealed near-complete resolution of the lesion with mild residual scarring and traction bronchiectasis (Figure The patient then underwent a CT-guided percutaneous core biopsy of the right middle lobe. Pathologic examination of this specimen revealed granulomatous and eosinophilic pneumonitis with rare fungal forms identified. Subsequently, the patient’s bronchial wash culture demonstrated fungal growth with s Figure .Curvularia species of fungi are ubiquitous and mostly constitute plant or soil pathogens found in tropical and subtropical climates. To date, more than 10 unique species have proven to be pathogens of clinical significance. Documented human mycoses due to Curvularia infection include onychomycosis, endocarditis, invasive sinusitis, keratitis, systemic infection, and cerebral phaeohyphomycosis. Generally speaking, phaeohyphomycosis is an infection caused by dematiaceous molds, of which Curvularia is one of more than 150 species pathogenic in humans [Curvularia have been associated with either an immunocompromised host or those with significant underlying lung pathologies such as bronchiectasis or cystic fibrosis. Hence, this case was highly unusual given the patient's lack of prior medical history, immunocompetent state, and the absence of any underlying lung pathology. It is important to note that infections can be seen adjacent to neoplasms in suspected lung malignancy. Diagnosis is very challenging as the primary mode of diagnosis for all agents of phaeohyphomycosis is fungal culture and histopathologic analysis of tissue aided by Fontana-Masson stain. However, even with these modalities, histopathologic interpretation can be difficult as different pathologic features can be produced in tissues by the same fungus due to suspected host factors [Curvularia species isolated in our case was susceptible to amphotericin, itraconazole, and posaconazole but resistant to voriconazole.n humans . However factors . ManagemCurvularia pneumonia in immunocompetent patients is very rare and the underlying risk factors predisposing to these infections are still unclear. We presented a rare case of phaeohyphomycosis pneumonia presenting as a unilateral mass with complete resolution after identifying the rare pathogen and providing susceptibility-guided treatment.


A 45‐year‐old man underwent esophagogastroduodenoscopy because of symptoms of laryngopharyngeal discomfort. We found a protruded reddish lesion adjacent to the ectopic gastric mucosa (EGM) in the cervical esophagus, and a biopsy revealed that it was a tubular adenocarcinoma. We diagnosed the patient with intramucosal cancer and performed endoscopic submucosal dissection. Esophageal endoscopic submucosal dissection was performed under general anesthesia using a conventional procedure. The resected tumor measured 23 × 14 mm and was adjacent to the EGM. Histologically, the tumor cells showed moderately well‐differentiated adenocarcinoma confined to the muscularis mucosa with no lymphovascular infiltration. Immunohistochemically, the tumor cells were positive for intestinal markers, namely MUC2 and CD10, and negative for gastric markers, namely MUC5AC and MUC6. The patient had no post‐endoscopy submucosal dissection stenosis and remained disease‐free without local recurrence. EGM of the cervical esophagus develops from the columnar epithelium during embryonic development. There are few reports on endoscopic submucosal dissection for mucosal cancer. Of these, immunostaining was performed in three cases. All were positive for MUC5AC and MUC6 and negative for MUC2 and CD10. Usually, EGM shows gastric type epithelium, but occasional cases with intestinal metaplasia, which show positivity for MUC2 and CD10, have been reported. Therefore, we consider this to be an extremely rare case of esophageal adenocarcinoma arising from intestinal metaplasia within the EGM. Ectopic gastric mucosa (EGM) of the cervical esophagus develops from columnar epithelium during the embryonic period,A 45‐year‐old man underwent esophagogastroduodenoscopy (EGD) because of symptoms of laryngopharyngeal discomfort. We found a protruded reddish lesion adjacent to the EGM in the cervical esophagus . The surface of the tumor showed a glandular and villous‐like structure, and narrow‐band imaging endoscopy showed an irregular microsurface and microvessel pattern  to accurately confirm the tumor margins, and ESD was performed using a single‐channel endoscope with a water jet  with a high‐frequency power supply unit  for electrocoagulation. After marking around the tumor, we started a mucosal incision using a dual knife  and performed submucosal dissection using an insulated‐tip knife nano  and a dual knife Figure . At presEsophageal adenocarcinoma developing within the EGM is extremely rare, and there have only been a few case reports of ESD performed for mucosal cancer.Helicobacter pylori infection, it has been reported that H. pylori were not found in the EGM of patients with H. pylori‐negative atrophic gastritis.H. pylori infection were found (data not shown), and other factors may be involved in intestinal metaplasia of the EGM. There are no reports of esophageal adenocarcinoma arising from intestinal metaplasia within the EGM; therefore, this case is extremely rare and gives valuable insight into the development of esophageal adenocarcinoma of the intestinal type.This case was positive for immunostaining of the intestinal type not only in the tumor but also in the surrounding epithelium. Therefore, we believe that the tumor developed from intestinal metaplasia. There are no reports that directly mention the cause of intestinal metaplasia in EGM. Although the involvement of bile acids has been reported in Barrett's epithelium,The authors declare that they have no conflict of interest.None.


Background: An extremely rare manifestation of perigraft seroma (PGS), in which a dense, semisolid jelly-like mass had formed around the shunt instead of the standard fluid-like form of the usual seroma, leading to misdiagnosis with other entities, such as tumors around the synthetic arterio-venous shunt (AVS) was presented.Case Report: A 64-year-old male with multiple myeloma post autologous bone marrow transplant with a renal impairment, presented with a rare form of PGS, which was noticed 2 months after placing a synthetic AVS vascular graft. The mass increased in size, and multiple attempts for excision failed due to recurrence, which led to tumor misdiagnosis. The mass reoccurrence stopped completely only after the radical shunt removal.Conclusion: This case report revealed a rare form of PGS, in which the seroma was represented as a firm, semisolid jelly-like mass rather than the typical fluid type transudate seroma. Despite its rarity, it was associated with a high recurrence rate because unlike the standard perishunt seroma, this semisolid jelly-like material could neither be aspirated, nor could it be resected en-bloc, leading to shunting dysfunction. Its management included advanced imaging and a high probability of shunt removal or replacement. Perishunt seroma or perigraft seroma (PGS) is an unusual but often devastating complication of vascular surgery. This disease is manifested by a persistent, often enlarging, sterile fluid collection confined within a fibrous pseudo-membrane around a prosthetic graft.Although such complications have been observed for several years, data regarding PGS are scarce, and information on this pathology is limited. Additionally, the majority of the literature is based on case reports, and no direct cause-effect or association has been established yet. Given its ultrafiltrated serum-like content and surrounding soft tissue capsule, a clear etiology is yet to be determined.PGS has been linked to Blalock–Taussig shunts, synthetic dialysis catheters, and arterial bypass surgery. It has been identified as a persistent sterile fluid collection confined by a fibrous capsule mostly in the mid-to-distal portion of the graft with an incidence rate of 1.7% to 4%.Regardless of its unclear etiology, PGS is considered one of the most serious complications of polytetrafluoroethylene vascular grafts, resulting in dysfunction and thrombosis.PGS can be clinically detected up to one year after insertion, with the highest incidence rate occurring during the first postoperative month after insertion, as reported in 25% of cases.Multiple hypotheses, ranging from graft handling and anastomosis to host cellular response, hemodynamic factors, and frequent puncture-related trauma have been proposed as possible causes of seroma, but none have been proven scientifically.The PGS gross pathology is usually composed of a glistening pseudocapsule encasing a sterile serous fluid collection around a prosthetic graft. Regarding the histology, it is mainly composed of mature fibroblasts with a rich collagen matrix forming the pseudocapsule and immature fibroblasts lining the outer graft with abundant chronic inflammatory cells, which are mostly giant multinucleated cells.Herein, a rare form of semisolid jelly-like component (rare variant of PGS) that necessitated the radical excision of the synthetic graft is presented. Documenting such rare presentations will raise the awareness for the differential diagnosis of non-vascular masses around synthetic shunts.A 64-year-old male with hypothyroidism was diagnosed with light-chain multiple myeloma in 2003 and underwent multiple external beam radiation therapy sessions, followed by two cycles of vincristine 0.4 mg/adriamycin 18 mg/dexamethasone 40 mg and zoledronic acid 4 mg.In December 2003, the patient underwent an autologous bone marrow transplant using the Melphalan protocol, which was complicated by graft-versus-host disease, which was managed with multiple lines of antimicrobial agents and corticosteroids.nd, 5th, and 6th thoracic vertebrae and cord compression at the tbl6 level, for which he underwent decompression and fixation followed by external beam radiation therapy.The patient was kept on regular follow-up until 2011, when he was found to have lytic lesions at the 2® Vascular Graft, W. L. Gore & Associates, Inc. Flagstaff, Arizona, USA) on the right forearm. The patient noticed a painless mass around the AVS on the right forearm almost 2 months later. The mass increased in size gradually until it reached 5 cm in diameter but without causing any symptoms other than some technical difficulties in accessing the shunt for hemodialysis. The mass was investigated by ultrasound, and it was reported as a non-vascular mass without any compromise to the venous or shunt systems. Therefore, an open biopsy was scheduled in June 2018.In 2018, the patient was diagnosed with renal failure in Palestine and underwent left forearm antecubital arterio-venous shunt (AVS) procedure to start hemodialysis. Subsequently, the shunt was thrombosed, and another treatment was performed to implant the synthetic AVS  replacement, Alternative therapies that have been tested in a small number of cases include replacement with homografts, native vein, saphenous vein wrapping, interposition of covered stents, and microfibrillar collagen injection into the periprosthetic space.Pseudocapsule removal and surgical drainage should be avoided because they are not curative and cause a high rate of infection.Plasmapheresis and intravenous fibrinogen administration have been used to eliminate hypothetical serum factors, which are thought to alter normal graft permeability.More recent animal studies revealed that radiation-crosslinked gelatin hydrogel of the prosthetic vascular graft can mimic the chemical and physical properties of the extracellular matrix in vivo, may aid in wound healing, and may effectively prevent serious sequelae, such as seroma formation and infection.In this case study patient, multiple surgical excisions were deemed unsuccessful due to technical challenges encountered in the removal of the semisolid jelly-like tissue, leading to the rapid recurrence. All the findings indicated the need for radical excision of the shunt to achieve resolution.Finally, exemption should be applied to sarcomas in solid non-vascular masses around AVS, notably epithelioid angiosarcoma, where almost all malignant endothelial cells are epithelioid in appearance. A total of 54% of the angiosarcomas arising from dialysis AV fistulas have the histological features of epithelioid angiosarcomas.Non-vascular masses around synthetic AVS in dialysis patients are considered a challenging entity, and can be serious and lead to shunt dysfunction. Although the differential diagnosis can be shortlisted with the histopathology of the biopsy, the findings can still be misleading due to the rare encounter of this semisolid jelly mass. Preservation of the shunt may not be an option to avoid recurrence.PGS is a rare presentation for a very common procedure . The most successful method in handling recurrent PGSs is the surgical excision of the AVS. This method has been proven to be the most effective, specifically in rare semisolid jelly-like types as presented in our case report. Documenting such rare presentations will raise the awareness for the differential diagnosis of non-vascular masses around synthetic shunts.Ethical approval and consent to participate:NAWritten informed consent was obtained from the patient‘s next of kin for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.NANAThe authors declare that they have no competing interests.NA


Congenital lobar overinflation (CLO) is a rare congenital pulmonary abnormality characterized by progressive overexpansion of a lobe(s) exerting mass effect on the remaining lobes and the mediastinum. Bilateral involvement is extremely rare and has been reported only a handful of times. We present a case of bilateral congenital lobar overinflation in a term female neonate managed with single-step bilateral lobectomies. CLO is a developmental pulmonary abnormality characterized by progressive overinflation of one or more lobes of the lungs with resultant mass effect and compression of the remaining lobes We report a rare case of bilateral CLO involving the left upper lobe and right middle lobe in a female newborn presenting with respiratory distress since birth. She was successfully managed with single-stage bilateral lobectomies. As to our search, this is the first reported case of bilateral CLO in Ethiopia.We report a case of a 28 days old female neonate who was born at a gestational age of 39 weeks and 4 days by caesarian section for an indication of 2 previous CS scars. She presented with fast breathing since 6 hours of age which progressively worsened over time. At presentation, she was tachypneic (65 breaths per minute). She also had subcostal/intercostal retractions and mild chest in drawing. Chest auscultation at initial presentation was unremarkable. Baseline investigations including CBC, RBS, RFT and serum electrolytes were normal except for a mild hypokalemia which was corrected. Blood culture was non-revealing. Abdominal ultrasound was also normal. Transthoracic echocardiography (not shown) revealed patent foramen ovale with left to right shunt. She was admitted with a working diagnosis of EONS. She failed to improve despite being put on IV antibiotics and intranasal oxygen. Chest radiograph and subsequently chest CT were obtained. The initial chest radiograph  revealedThe chest CT obtained at the age of 28 days  revealedThe diagnosis of impending respiratory failure secondary to bilateral congenital lobar overinflation was made, and the infant was prepared for surgery. Thoracotomy and single-stage bilateral lobectomy of the left upper lobe and right middle lobe was performed. She had a smooth postoperative course. A chest radiograph obtained on her seventh postoperative day  showed oCongenital lobar overinflation, also known as congenital lobar emphysema is an uncommon abnormality of the lungs characterized by overinflation of a lobe. It is thought to be due to an intrinsic or extrinsic bronchial abnormality causing a ball-valve mechanism with resultant hyperinflation of the affected lobe Imaging plays an important role in the diagnosis of CLO. Radiographs obtained soon after birth may show opacity of the involved lobe due to retained fetal fluid. Subsequent films will demonstrate hyperlucency and hyper expansion of the affected lobe with associated mediastinal shift and compression of the remaining lobes. CT will better demonstrate the extent of involvement and degree of mass effect. CLO appears as a hyperexpanded lobe with attenuated pulmonary vasculature. CT will also exclude secondary causes of lobar overinflation such as compressing mediastinal masses and vascular anomlies The management of bilateral CLO has been a topic of debate. There are 2 major schools of thought. The first one involves performing a staged surgery with removal of one of the affected lobes . The child would then be followed and a second surgery to remove the other lobe would be done if indicated. If the child remains stable and there is no significant rebound hyperinflation of the non-removed diseased lung, conservative management would be sought. The rationale behind this approach is that a single stage bilateral lobectomy is a high-risk procedure with increased risk of complications and postoperative pain. In the reviewed case reports and case series, this approach appears to be the more popular one. In the largest single institution case series on bilateral CLO, 3 of the 4 patients were managed with unilateral lobectomy. They were followed and remained stable without needing a second surgery In conclusion, bilateral congenital lobar overinflation is an extremely rare cause of respiratory distress in the newborn. Its diagnosis requires high index of suspicion and relies on imaging, typically with chest radiographs and CT. There are 2 main approaches to its management including single step bilateral lobectomy and staged (2-step) surgery.