The locally advanced pancreatic cancer has been steadily recognized as a potentially curable disease by a combination of chemotherapy and surgery. The remarkable effect of advanced chemotherapy would help surgeons do a function-preserving operation for advanced pancreatic cancer.2 of gemcitabine and 125 mg/m2 of albumin-bound paclitaxel, the tumor shrunk based on imaging done every four months during chemotherapy, with residual periarterial high-density area around CA and proximal SMA and the patient was referred for surgery. During the operation, the absence of cancer cells was confirmed at (1) the origin of the proper hepatic artery, gastroduodenal artery and the left gastric artery, and (2) pancreatic cut stump along the right border of the portal vein; thus, distal pancreatectomy with coeliac axis resection was done. The patient had postoperative adjuvant chemotherapy with 100 mg/day of tegafur/gimeracil/oteracil for half a year and is currently alive and well, without signs of recurrence and diabetes mellitus a year after surgery.A 73-year-old woman presenting with obstructive jaundice was diagnosed to have a 3-cm pancreatic body cancer invading the celiac axis (CA), superior mesenteric artery (SMA), portal/splenic vein confluence, and the common bile duct (CBD). A plastic internal stent tube was placed endoscopically. After 11 cycles (231 days) of a weekly doublet chemotherapy with 1000 mg/mAlthough surgical techniques aimed at local radicality are important, especially for conversion surgery for locally advanced pancreatic cancer, surgeons should consider the balance between radicality, safety, and functional preservation of surgery. •A locally advanced pancreatic cancer has been curable by chemotherapy and surgery.•The arterial invasion by imaging diagnostics is sometimes released by chemotherapy.•The common bile duct invasion was dissolved after chemotherapy.•The effect of chemotherapy would help surgeons do a function-preserving operation. Due to abnormal liver enzyme levels based on blood chemistry, the patient was referred to our hospital. The patient was 146 cm in height and 49.8 kg in weight, and three months prior to visit to our hospital, she had had no body weight loss. She had no comorbidities, except for hyperlipidemia for over 13 years, and she never drunk nor smoked. The blood chemistry at initial hospital visit showed elevated levels of serum total bilirubin , aspartate aminotransferase , alanine aminotransferase , and alkaline phosphatase . The fasting blood glucose of the patient was slightly elevated , while the HbA1c level was normal . The serum levels of pancreatic cancer tumor markers were all within normal range, as CA19–9 was 24.2 U/mL [<37], CEA was 2.3 ng/mL [<5.0], DUPAN-2 was 95 U/mL [≤150], and SPan-1 was 23.2 U/mL [≤30].An abdominal computed tomography (CT) scan with dynamic enhancement revealed a 3-cm, ill-defined tumor in the pancreatic body circumferentially invading the CA and SMA a,c,d,e. 2 of gemcitabine and 125 mg/m2 of albumin-bound paclitaxel for three to four weeks was started 40 days after the first visit to our hospital. Because the patient suffered from grade 4 neutropenia after the first two administrations during the first cycle of the doublet regimen, the dose was set at 70% of full dosage and was administered two to three weeks. The effect of chemotherapy was estimated by a monthly evaluation of tumor markers and CT scan every four months. The tumor shrunk on each imaging evaluation during chemotherapy, with residual periarterial high-density area around the CA and proximal SMA  the origin of the proper hepatic artery and gastroduodenal artery and (2) the origin of the left gastric artery. The pancreas was cut along the right border of the PV, and there were no cancer cells with patchy deciduation of exocrine tissues and fibrous tissue proliferation at the cut surface; thus, DP-CAR, preserving the left gastric artery, was selected asss a preferable procedure The gross examination of the surgical specimen revealed 43 × 22 × 14-mm, ill-defined, pancreatic body tumor with an irregular whitish area and loss of lobulation. Microscopically, the tumor was a moderately differentiated tubular adenocarcinoma, encroaching beyond the pancreatic capsule but showing absence of invasion to the adventitia of resected CA and PV; thus, it was assigned as ypT3, ypN2 (four metastases out of 50 harvested nodes), ypM0, ypStage III according to the 8th edition of TNM classification. The cancer cells were observed very near to but more than 1 mm from the cut surface.The patient had a postoperative adjuvant chemotherapy with 100 mg / day of S-1® (Tegafur/gimeracil/oteracil) for half a year and is currently alive and well, without signs of recurrence and diabetes mellitus a year after surgery.3Owing to increasing safety with mortality rates under 5% The expected effects of chemotherapy for LAPC prior to resection are (1) to provide a patient selection by testing the chemoresponsiveness of the tumor to identify aggressive diseases and to spare ineffective therapies and (2) to increase the complete resection rate by shrinking the tumor and treating micrometastases. As for surgical procedures after chemotherapy, almost all patients with LAPC with more than 180 degree of invasion to the CA and/or SMA spared combined resection of such major arteries Recently, FOLFIRINOX has been the major regimen for the neoadjuvant treatment of LAPC with high rate of conversion Surgical procedures have been continuously refined and have markedly improved the survival outcomes of patients with pancreatic cancer, in combination with more effective adjuvant chemotherapy regimens 4Herein, we report a case of a patient with pancreatic body cancer who presented with obstructive jaundice and spared total pancreatectomy owing to the marked effects of preoperative chemotherapy. Although surgical techniques aimed at local radicality are important especially for the conversion surgery for LAPC, surgeons should progressively consider the balance between the radicality, safety, and functional preservation of surgeries.Not applicable as per institutional policy.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.The data that support the findings of this study are available from the corresponding author, [JY], upon reasonable request.All authors received no financial compensation related to the development of the manuscript.N/A.Junji YamamotoNot commissioned, externally peer-reviewed.HK drafted the manuscript and JY and HK participated in treating the patients and revised the manuscript. JY, MH, and YK participated in the surgery and postoperative management. TI investigated these cases pathologically. All authors read and approved the final manuscript.All authors have no affiliations with or involvement in any organization or entity with any financial interest, or non-financial interest in the subject matter or materials discussed in this manuscript.


Total or partial vulvo-vaginectomy or vaginectomy are not routinely surgery due to the complexity of the techniques and because they are considered radical treatments. Furthermore, in literature, there is a paucity of information regarding these techniques and the extent of the reproductive tract resection is not always clearly defined, as the same technique is often named in a different way by different authors, confusing the reader. The aim of this article is to review and describe five surgical procedures on the basis of the correct identification of the anatomical areas: partial vaginectomy, complete vaginectomy, partial vestibule-vaginectomy, vulvo-vestibule-vaginectomy and vulvo-vestibulectomy. For each technique, indications and possible intraoperative and perioperative complications are mentioned. Moreover, authors’ clinical experience in 33 dogs presenting genitourinary lesions not amenable to local resection via simple episiotomy and outcomes are described.Total or partial vulvo-vaginectomy or vaginectomy are not routinely performed due to the complexity of the techniques and because they are considered radical treatments. Little information can be found in the literature, as the same technique is often named in a different way by different authors, confusing the reader. Therefore, the aim of this essay is to describe five different surgical techniques: partial vaginectomy, complete vaginectomy, partial vestibule-vaginectomy, vulvo-vestibule-vaginectomy and vulvo-vestibulectomy. All techniques are described on the basis of the correct identification of the anatomical nomenclature related to structures involved in surgery, in order to give a more precise and unambiguous description and execution of surgical techniques. Moreover, possible intraoperative and perioperative complications and the authors’ clinical experience in 33 dogs are described. All techniques are well tolerated and could be curative in case of benign or malignant tumours that have not yet metastasized and palliative in other cases. Moreover, they are also useful for therapeutic purposes for chronic vaginitis, severe vaginal cysts or congenital abnormalities. It is our opinion that having five different available techniques to approach vaginal disease is useful to perform the best surgery according to the clinical findings, patient’s characteristics, technique invasiveness and whether it is palliative or not. Many diseases, such as benign or malignant neoplasia, vaginitis, vaginal prolapse and congenital abnormalities, could affect the copulatory organs  in the dog and most of them could be age-related . For theUnfortunately, in literature there is little information regarding the surgical techniques performed on the copulatory organs. In particular, the extent of the reproductive tract resection is not always clearly defined, as the same technique is often named in a different way by different authors, confusing the reader.In the authors’ opinion, a correct anatomical nomenclature related to structures involved in surgery could lead to a more precise and unambiguous description and execution of surgical techniques.Therefore, our purpose is to give a more specific and specialized point of view, starting from the Nomina Anatomica Veterinaria (N.A.V.) to describe in detail five different surgical techniques available for the female reproductive system of the dog.N.A.V. subdivides the female reproductive system in Organa genitalia feminina  and Partes genitales femininae externae  . The vagOn the basis of the correct identification of the anatomical areas, five surgical techniques can be described: partial vaginectomy, complete vaginectomy, partial vestibule-vaginectomy, vulvo-vestibule-vaginectomy and vulvo-vestibulectomy .Partial vaginectomy involves the removal of a variable tract of the vagina, always including the cervix. All vaginal tissue, from the cervix to the hymen, are removed in complete vaginectomy. A partial vestibule-vaginectomy is similar to the complete vaginectomy, but a variable part of the vestibule is involved: in this case, a urethrostomy performed in the segment of the vestibule tissue left in situ is mandatory. In a vulvo-vestibule-vaginectomy, all the copulatory organs  are removed and a perineal urethrostomy has to be performed. In a vulvo-vestibulectomy, the externa genitalia and a variable vestibular length is removed: the urethral tubercle is not involved . Except The objective of this study is to describe, in function of the N.A.V. organs’ description, these five different surgical approaches to the bitch reproductive system as performed by the authors. Since the literature provides scarce and ambiguous explanation about these kinds of techniques, another purpose is to offer to practitioners an exhaustive description in order to provide a repeatable, safe and tolerable surgical procedure. For each technique, indication and possible intraoperative and perioperative complications are mentioned. Moreover, authors’ clinical experiences in 33 female dogs presenting genitourinary lesions not amenable to local resection via simple episiotomy and outcomes are described.In the next paragraph, partial vaginectomy, complete vaginectomy, partial vestibule-vaginectomy, vulvo-vestibule-vaginectomy and vulvo-vestibulectomy, as performed by the authors, will be described. All information described below is summarized in Partial vaginectomy (PV) is requested when the first third of the vagina is involved in a pathological process. The main indication for a partial vaginectomy is the treatment of benign or malignant neoplasia, chronic cranial vaginitis unresponsive to medical or surgical treatment such as urethropexy, or cervix trauma occurred during parturition. In the case of malignant tumour, PV is recommended if there is no local dissemination or distant metastases. It could also be performed as a palliative procedure for advanced malignancies when the owner does not allow other surgical procedures, the tumour pushes organs such as rectum or bladder, or in elderly patients. Chronic cranial vaginitis could be consequent to congenital malformation of the vagina and vestibule that cause an urinary reflux or to an abnormal outflow of urine or urine stagnation in the vagina caused by topographic abnormalities of the genital tract following sterilization at a very young age .PV is performed via an abdominal approach with the patient in dorsal recumbency. A ventral midline incision from the umbilicus to the pubis is performed. In intact or ovariectomized dogs the surgical procedure is completed in two steps: in the first step, routine ovariohysterectomy (OVH) or hysterectomy are performed and the bladder is retroflexed to expose the vagina .In the second step, the cranial vagina is resected. The rectogenital and vesicogenital pouch are carefully dissected via blunt and sharp dissection with the purpose to allow the complete isolation of the vagina. The dissection must be performed very close to the vagina in order to prevent inadvertent trauma to the ureters, and to preserve the innervation of the bladder and urethra. Vascular supply  that runs laterally the cranial part of the vagina are ligated close to the vagina and close to the rectogenital and vesicogenital pouch. The vagina is then cranially pulled to allow the resection as caudally as possible. The vaginal stump is sutured with single, introflexing detached sutures. The abdominal wall is then closed as routine.PV performed via abdominal approach presents minimal complications, but the surgeon must be careful when working between the two ureters at the base of the broad ligament lateral the uterus body and vagina: in overweight or obese dogs, the ureters could be difficult to recognize. Urethra must be recognized and not damaged as well as vaginal vascularization to avoid postsurgical complications. The use of a urinary catheter makes it easier to recognize the urethra. Moreover, in some cases, it could be necessary to resect the vagina more caudally, depending on the extension of the lesion: in this case it could be necessary to tie the vaginal artery and vein. These vessels must be ligated as close as possible to the vagina in order to avoid unintentionally tiding of bladder and urethra vascularization.However, depending on the breed, the patient state of nutrition, neoplasia location or topographic position of the vagina, it could be necessary to break through the vesicogenital pouch that divided the peritoneal cavity from the retroperitoneal one in order to allow the complete resection of the organ involved. Moreover, the uterine and vaginal artery  on the lateral wall of the vagina, the pelvic plexus  located on both side of the cervix and pudendal nerve that runs along the dorsal edge of the internal pudendal artery must be identified and preserved. It must be emphasized that in PV, due to the pubic bone, a complete visualization of the structures in this area could not be always possible, thus palpation could help their identification.Intraoperative complications are haemorrhage, urethral damage (such as rupture or stretching) and lesion of the rectum, bladder or clitoris’ corpora cavernosa consequent to a damage of the pelvic plexus. In the post-operative period, it is sometimes possible to detect urethral spasms that could led to transitory urinary retention more frequently than urinary incontinence. Intra- and post-operative complication could be avoided or minimized if the surgery is properly performed as described and all steps are carefully executed.Complete vaginectomy (CV) is indicated when benign or malign tumours, chronic vaginitis or neoformations involving the entire vagina and a PV cannot be performed.First of all, ovariohysterectomy (OVH) or hysterectomy via abdominal approach are performed in intact and ovariectomized dogs. If the dog has previously undergone ovariohysterectomy, before CV is performed, it may be necessary to solve possible adhesions between the vaginal stump, the bladder and the rectogenital and vesicogenital pouch.CV is performed via a perineal approach with the dog in sternal recumbency. The perineal skin, from the base of the tail to the vulvar lips and laterally to the mid-thigh region, are shaved. A pursue-string suture is placed in the anus and a sterile Foley catheter is inserted in the urethra. Immediately after surgery, the pursue-string suture is removed. A midline incision between anus and the dorsal commissure of the labia of the vulva  is made. Tissue and muscle  are carefully dissected via blunt and sharp dissection in order to allow the complete isolation of the vagina. Vascular supply to the vagina  is ligated as close as possible to the organ in order to prevent damage to the other vessel or nerve that could be found in this anatomical region. The isolated vagina is then retroflexed and retracted caudally. Attention must be paid to the urinary meatus, which must not be removed or damaged during the resection of the vagina: the urethra and its junction with the vaginal floor are identified via the urinary catheter and the vagina is resected just cranially in order to left enough tissue to perform an introflexing detached suture. That allowed the complete asportation of the vagina and hymen without involving the urinary meatus. The subcutaneous and cutaneous tissue are closed with attention to ureters, urethra, vessel and pelvic plexus which must not be involved in the suture. Intra- and post-operative complication are similar to those described for PV.Partial vestibule-vaginectomy (PVV) is recommended as treatment for benign or malignant neoplasia, chronic cranial vaginitis unresponsive to medical or surgical treatment, congenital and acquired malformation like stenosis, cyst or paramesonephric residues, vaginal diseases near to the hymen or in the proximal third of the vestibule.Ovariohysterectomy (OVH) or hysterectomy via abdominal approach are previously performed in intact and ovariectomized dog.PVV could be performed by perivaginal approach with the dog in sternal recumbency. The surgery procedure is the same as previously described for CV, but the resection of the organ includes, besides the vagina, a variable part of the vestibule depending on the extension of the lesion.During this procedure the urethra is identified via the urinary catheter, dissected free via blunt dissection and resected at its junction with the vestibula floor. After the resection of the vagina and vestibule, dead spaces are closed by reopposing adjacent soft tissue structures. In order to perform the urethrostomy, an episiotomy is needed to expose the floor of the remaining vestibule. An incision on the midline of the vestibule floor is then performed, the spatulate cut end of the urethra is exteriorized through the small stab incision and the urethral mucosa is sutured to the vestibule mucosa with a 4-0 absorbable monofilament glycomer simple interrupted suture. As the urethrostomy is completed, the episiotomy incision will be closed with a simple continuous pattern with a braided polyglycolic acid absorbable suture in three layers: vestibula mucosa, muscles and subcutaneous tissue and lastly the skin.Intra- and peri-operative complication are the same as described for the other surgeries. A Foley catheter must be left in situ for a couple of days to allow complete healing of the urethrostomy.Vulvo-vestibulo-vaginectomy (VVV) is indicated for benign large or multiple neoplasia involving the vagina, vulva and vestibule, malignant neoplasia, chronical vaginitis and vestibulitis.VVV could be performed by a mixed approach  or caudal approach alone: a perineal urethrostomy is always required.The mixed approach is mandatory in intact or ovariectomized dog and in all cases with suspected abdominal adhesion. The surgical procedure is completed in two steps: an abdominal approach for ovariohysterectomy (OVH) or hysterectomy and a caudal approach for the resection of the vagina, vulva and vestibule. The surgical field is prepared as described for the perineal approach for the CV procedure.Subsequently to OVH or hysterectomy, the bladder is retroflexed to expose the vagina and associated structures. The surgical procedure is the same as described in the PV. In the second step, the patient is placed in sternal recumbency to expose the perineal region. An elliptical incision around the vulvar margin is made, followed by the clitoral artery isolation and ligation in order to prevent emorrhagia. The caudal vagina and vestibule are freed via blunt dissection from the surrounding tissue and ischiocavernosus, ischiourethralis, the superficial branch of the external sphincter ani muscle and constrictor vestibuli muscles are resected, in order to allow a complete dissection . The vagIn caudal approach all the vagina, vestibule and vulva are completely isolated from the surrounding tissue and resected as described above. The vaginal artery must be ligated as deeply as possible.In both caudal and mixed approach, the urethra must be managed with caution to prevent damage to the distal tract and must be freed from the muscular component to correctly perform the urethrostomy. If the vessel of vaginal and clitoral are not correctly identified and ligated, a haemorrhage may occur.Vulvo-vestibulectomy (VV) is indicated for benign clitoral neoformation, malignant not infiltrated neoplasia, vulvo-vestibular traumatic or congenital lesions such as labial aplasia or vulvar stenosis.The patient must be prepared as described for the caudal approach of the CV. An elliptical incision around the vulva is performed, followed by the clitoral artery isolation and ligation. Vulva and vestibule are dissected via blunt and sharp dissection from the surrounding tissue and the ischiocavernosus, ischiourethralis and superficial branch of the external sphincter ani muscles are resected in order to allow vestibula resection just caudally of the urinary meatus. Subsequentially, a restriction of the remnant part of the vestibula diameter, the stump of the genital tract will be pulled caudally and inclined in order to prevent an incorrect urinary out flow or vaginal urinary stagnation: the vestibule mucosa is then sutured to the skin with simple interrupted suture.The technique does not require particular attention, except to avoid injuring urethra and urinary meatus.The medical records of thirty-three female dogs that underwent PV, CV, PVV, VVV or VV from 2008 to 2021 were reviewed. Age, breed, medical history, clinical findings, preoperative diagnostics, post operative management, complications occurred during the surgery and in the postoperative period, and histological diagnoses were recorded.In all cases where a vaginal mass was suspected, diagnosis has been made on the basis of history and clinical evaluation that include vaginal and rectal palpation: in some cases, biopsy or fine-needle cytology of the mass were performed. Ultrasonography of the reproduction tract , biochemical and haematologic profiles, abdominal ultrasound examination  and thoracic radiographic  for cancer staging (when necessary), vaginal cytology was performed in all bitches.®, Ati PETS, Bologna, Italy) or 0.005 mg/kg dexmedetomidina  and 0.2 mg/kg metadone , general anaesthesia was induced by administration of 2–4 mg/kg propofol  intravenously. Anaesthesia was maintained with 1.5–2% isoflurane  in oxygen.In each bitch, after a premedication with an intramuscular injection of 0.01–0.02 mg/kg acepromazina  and the mean age was 9.39 years sd 2.96 . Breeds represented included mongrel (12), Beagle (1), Boxer (3), Labrador (2), Yorkshire Terrier (2), Cavalier King Charles (1), Cirneco dell’Etna (1), Dalmatia (1), English Bulldog (1), English Setter (1) English Cocker Spaniel (1), French Bulldog (1), German Shepherd (1), Golden Retriever (1), Irish Setter (1), Maremman Shepherd (1), Pointer (1) and Siberian Husky (1). Signalment data of the patients are reported in At presentation, as reported in Dog 26 arrived to our clinic for an uroperitoneum originated from a vaginal rupture following surgical closure of the urinary meatus following the surgical removal of a vaginal mass.The surgical technique was planned in function of the clinical examination, abdominal ultrasonography, vaginoscopy and/or vaginography performed and the direct intraoperative visualization of the neoplasia and/or cytological/biopsy preoperative evaluation.Thirty dogs underwent surgery for removal of a vaginal or vulvar neoformation, two for a fluid filled vagina, and one for a fluid filled vagina associated to a chronic vaginitis. PV was performed in 14 dogs, CV in one dog, PVV with a vestibule urethrostomy in two dogs, vulvo-vestibule-vaginectomy with a perineal urethrotomy in seven dogs and VV in nine dogs. Ovariohysterectomy was performed in all intact dogs that underwent PV or CV and VVV. In two dogs who had undergone, respectively, PVV and PV and were previously neutered by ovariectomy, hysterectomy was performed.Surgery was successfully achieved and well tolerated in all dogs.®, Formevet, Milano, Italy) and 2–4 mg/kg carprofen  or 1 mg/kg robenacoxib  once a day for 3 days to reduce inflammation and edema. Amoxycillin/clavulanic acid  at 12.5 mg/kg twice a day for 10 days was administered as prophylaxis.An urinary catheter was left in place for 24–48 h in VV and up to 7 days in VVV or PVV; a petroleum jelly was applied to the perineum to prevent urine scalding at least for 10 days. An Elizabethan collar was used until the removal of the suture. Pain was managed with the administration of 2–4 mg/kg tramadol every 8–12 h  was hospitalization longer. In dog 12, who underwent vulvo-vestibule-vaginectomy and ovariohysterectomy because of neoformations in cranial vagina and in the vulva, the surgery was quite long and a severe haemorragia was observed, requiring a longer recovery due to pain management and blood transfusion. In dog 26, the longer hospitalization was according to clinical alteration referred to the uroperitoneum presented before surgery. In all cases, except for two (dog 1 and 12), the short-term outcome of 7 days was without complications. No micturition or defecation problems were observed, nor ureters or bladder anomalies were ultrasonographically detected in post-operative stage. In dog 1, as a large skin excision lateral the urethrostomy area was necessary, the healing process led to a deviation of the urethrostomy site and a consequent deviation of the urine jet was observed.The postoperative management of dog 26 was without complication, even if in this case surgery was a palliative cure, as metastases were observed at the presentation.Twenty-eight samples were analysed via histopathology; in five cases, the owners refused the histological examination. Vaginal neoplasia (25/28), an intramural cyst (2/28) and a fluid filled vagina with a severe chronic purulent ulcerative vaginitis (1/28) were diagnosed. The results of the histological or cytological examination, performed during the preoperative evaluation, has been confirmed by the histopathological examination done after surgery excepted for dog 33 where the cytological examination was in doubt because the poor cellularity. The results of the histopathological examination are shown in Aim of this article was to describe five different surgical techniques to correct diseases affecting the vaginal, vestibular and vulvar tract in the bitch. To date, the literature reports only four surgical techniques involving this reproductive tract: the authors instead differentiate five techniques depending on the portion that has to be removed and gave instructions to plan the best and less demolishing intervention in agreement with the disease, the age and the physical condition of the patient. Of the five surgical techniques we have described, in the literature, no cases of vulvo-vestibulectomy  are reported. The surgical techniques depicted in this essay are classified in function of the anatomical female reproductive tract involved in the surgery as illustrated in The main problem we had to face, related to surgical techniques described in the literature, is linked to the nomenclature, to not very detailed description of different approaches in the literature, and to the indications relating to the sections of the apparatus that have to be removed. Due to these unclear aspects, it was not easy to understand what type of intervention was performed in the literature and correctly compare the techniques and the results obtained.Surgical textbooks , reviewsAs regards demographic aspect, our data are in line with what has been found in the literature: adult bitches (average age 9 years), mainly whole dogs with neoplastic concurrent diseases ,12,13,15In our cases, 13 of the 25 tumours analysed histologically were malignant, and 12 were benign. This result seems to be in contrast with data concerning the incidence of vaginal tumours in the literature as it was reported that benign tumours show a higher incidence than malignant tumours ,17,18: oThe 5 techniques described were well tolerated by the patients and a complete recovery and hospitalization was observed within 24–48 h in accordance with what is described in the literature ,14,15. PIn the post-operative period (short term outcome 7 days), we did not find any complications affecting the urinary tract and colon/rectum. Two cases of transient urinary incontinence (case 1 and 6) which resolved spontaneously in a couple of days were observed  and oneThe minor post-operative complications observed in our cases are in agreement with what is described in the literature ,14,16,19Ogden et al.  and NeliSome authors consider these very specialized and very invasive/radical surgical techniques and therefore advise against them ,4, whileAlthough CT scans were not performed, as the owners did not consent, it is the authors’ opinion that performing advanced diagnostic tests may be useful in order to better establish the margins of the lesion and the degree of the infiltration: however, in all 33 cases described in the article , surgery was 100% resolutive and the margins decided on the basis of the preoperative and intraoperative diagnostics were sufficient. In reality, among 44 cases described in the articles mentioned, only 12 cases underwent preoperative CT (1 case of  and 11 oIt is our opinion that having five different available techniques to approach vaginal neoplasms depending on their size, location and nature is useful in order to perform the best surgery according to the patient characteristics, technique invasiveness and whether it is palliative or not, as sometimes we had to improve only the quality of life, so it may be appropriate to choose a technique rather than another less invasive and destructive one.Moreover, in our opinion and experience, the possibility to perform an urethrostomy at the vestibular level instead of percutaneous urethrostomy presented several advantages in order to protect the urinary tract from infections or trauma. Finally, nowadays even in veterinary field, the importance of aesthetic factors must be considered, for example, when we leave the vulva.The main limitation of our study is related to the kind of surgical techniques described, which are rarely needed and performed in practice. Therefore, due to the number of patients, it was not possible to perform an adequate statistical analysis or identify particular predispositions within the study population.Another limitation is represented by the difficulty to compare our data with those previously published, as the literature reports little information and an ambiguous description of these surgical techniques.We described in detail five different surgical techniques available for the female copulatory organs related to a correct anatomical nomenclature. All techniques here described results well tolerated by the patient. In the authors’ opinion, the possibility to choose between five different techniques according to the portion of the female reproductive tract that has to be removed and the possibility to plan the best and less demolishing intervention in agreement with the disease, the age and the physical condition of the patient are important points.


Extranodal natural killer/T-cell lymphoma (ENKTL), nasal type, generally affects the orbit by direct extension. It can even rarely present as severe intraocular inflammation mimicking endophthalmitis. Delayed diagnosis and treatment are frequently reported.A 43-year-old woman presented with 2-month blurred vision in her left eye. Ocular examination revealed hypopyon panuveitis. She was initially diagnosed with endogenous endophthalmitis, which proved irresponsive to antimicrobial therapy. High-dose prednisolone was given afterward, but this failed to stop the development and continuous progression of ocular inflammation. The diagnosis of ENKTL was finally confirmed from the pathological findings of oral ulcers and cervical lymph nodes and chemotherapy was prescribed. After the first cycle of chemotherapy, the patient’s ocular inflammation subsided. Unfortunately, her left eye became phthisis with progressive visual loss within 9 months following the diagnosis.The diagnosis of intraocular ENKTL is frequently delayed, which can lead to severe problems for treatment as the disease is aggressive with a poor prognosis. It can be found not only in elderly patients but also in a middle-aged individual. In patients with ocular inflammation, a thorough systemic evaluation and histopathological examination of the associated systemic findings is extremely helpful and may reveal the cause of the ocular inflammation, including revealing possible ENKTL. Extranodal natural killer/T-cell lymphoma (ENKTL) is an extremely rare subtype of non-Hodgkin’s lymphoma (NHL), believed to be an Epstein–Barr virus-related neoplasm –3. The iA 43-year-old woman who presented with a 2-month painful blurred vision of her left eye was referred to our uveitis clinic with hypopyon panuveitis in the left eye. Ocular examination revealed a visual acuity (VA) of 6/6 in the right eye and counting fingers at 1-ft in the left eye, intraocular pressure was 12 and 6 mmHg. Her left eye showed 4+ anterior chamber cells with 1.7 mm of hypopyon and a 2+ to 3+ flare with only mild conjunctival injection. There were multiple iris nodules, as shown in Fig. Streptococcus pneumoniae grew from his eye discharge. The eviscerated globe revealed high-grade ENKTL [Our case study involved a rare intraocular presentation of UMS resulting from ENKTL. Its presentation can sometime lead to misdiagnosis and it can be difficult for general ophthalmologists to manage. The first case of intraocular ENKTL was reported by Maruyama et al. in 2015, and involved a 66-year-old female patient with a VA HM. She presented with dense vitreous opacity and vitreous clumping . Zhang ede ENKTL –12. Accode ENKTL . We are de ENKTL . Delayed


The cause of pelvic pain remains a significant diagnostic challenge, even for experienced radiologists. An accurate differential diagnosis has to be done according to the patient's age and gender. Spontaneous round ligament hematoma is an uncommon cause of acute pelvic pain in adult female patients. To the best of our knowledge, it has never been reported in the literature in the paediatric population. Ultrasound examination is the first line imaging modality for pelvic pain evaluation in young women but it might result inconclusive. Thanks to its panoramic view and multiparametric approach, the MRI can play a pivotal role in the diagnosis of spontaneous round ligament hematoma in paediatric female patients, resulting in a more effective patient's therapeutic management. Pelvic pain in paediatric population is one of the most common causes of admission to the hospital through the Emergency Department (ED) or Urgent Care (UC). It still represents a diagnostic challenge, even for experienced radiologists.An accurate differential diagnosis needs to be carried out according to the patient's age and gender. The differential diagnosis for pelvic pain in an adolescent female is broad, ranging from gastrointestinal  to genito-urinary  conditions Clinical examination, laboratory tests and imaging examinations are crucial to reach a proper diagnosis. A detailed reproductive history, specifically in young women patients presenting with pelvic pain, should also be taken into consideration Spontaneous round ligament hematoma is an extremely rare cause of acute pelvic pain in adult female patients Imaging plays a pivotal role in the diagnosis and the therapeutic management of this condition in paediatric female patients presenting acute pelvic pain.A 15-year-old girl was admitted at a different hospital reporting recurrent episodes of severe intermittent pain at the right iliac fossa that began three days prior.The young patient reported normal, painless, menstrual cycles, and her blood tests were unremarkable. No recent trauma nor sexual activity were reported.The patient performed a lower abdomen ultrasound which showed a large inhomogeneous hypo-anechoic mass (estimated diameter = 10 cm) in the right iliac fossa. Therefore, the patient was referred to our institution where she was scheduled for a contrast-enhanced MRI of the pelvis.2) DWI- weighted images  fluid collection, which was appearing hyperintense on T1w, T2w and high-b value  and provides, along with other ligaments and muscles of the pelvic floor, stability to this region and support of the pelvic viscera. Medical literature on the round ligament's bleeding remains poor In 1982, Borrero reported the case of a 43-year-old woman with a groin painful mass appearing on straining mimicking an incarcerated inguinal hernia More recently, Kim and colleagues in a retrospective study of 59 patients diagnosed with postpartum hemorrhage found that non-uterine arteries, including the arteries running within the round ligament, are the major sources of post-partum haemorrhage and, thus, an important target for embolization Some authors also reported cases of hemoperitoneum following the bleeding of Sampson's artery in patients with history of inguinal hernia repair Mine et al Round ligament can be also involved by endometriosis: in those cases, the round ligament appears thickened or nodular, with or without small internal T1w hyperintense haemorrhagic foci As far as we know, no spontaneous round ligament haematoma has been yet reported in paediatric female patients.A specific diagnosis of round ligament hematoma based on ultrasound examination is not possible: US can demonstrate an inhomogeneous cystic lesion containing debris occupying part of the pelvic cavity. Vermiform appendix and normal ovaries should be always identified when an US is performed in a female patient.2) DWI- weighted images, the hematoma shows a diffusion restriction due to the high viscosity related to the proteinaceous and haemoglobin degradation products’ content Thanks to its panoramic view and multiparametric approach, the MRI facilitates a better definition of the anatomical structures of the pelvis as well as the tissue characterization, which is based on signal intensities on different sequences. Furthermore, MRI has no radiation exposure. Hence, it represents the appropriate method for a correct diagnosis. On MRI, a recent round ligament hematoma appears hyperintense on T2w and on T1w images with and without fat suppression due to the paramagnetic effect of methaemoglobin. On high-b value , which dislocates the ovary otherwise normal in morphology and signal, should raise the suspicion of round ligament haematoma in case of no signs of deep pelvic endometriosis.Hence, surgical exploration and drainage might be needed.When dealing with female adolescents suffering from acute pelvic pain, in the absence of any other potential causes, spontaneous round ligament hematoma should be considered as a rare but potential diagnosis.US is the first line imaging modality in young female patients having pelvic pain, but it might result inconclusive. In such cases, MRI is a problem-solving technique that can guide an effective patient's treatment management.The patient provided a written informed consent for using anonymized data for publication.All procedures performed in the study were in accordance with the ethical standards of the institutional and/or national research committee and with the 1964 Helsinki declaration and its later amendments or comparable ethical standards. Consent to participate: not applicable for this type of the work (case report).


Tuberous sclerosis complex is an inherited multisystemic disorder with manifestations in various organ systems as a result of a mutation of 1 of 2 tumor suppressor genes, tuberous sclerosis complex-1 or tuberous sclerosis complex-2. Perivascular epithelioid cell tumors have been shown to be associated with these gene mutations and include a variety of tumors such as angiomyolipomas and lymphangioleiomyomatosis.In this report, we present a case of a 28-year-old woman presenting with symptoms of severe abdominal pain and nausea with a medical history of cardiac rhabdomyoma, adenoma sebaceum, Ash leaf spots, bilateral renal angiomyolipomas, and retinal hamartoma, which are manifestations of tuberous sclerosis complex. The patient was operated twice for colonic perforations in the rectosigmoid and ileocecal regions where the pathologic examination revealed multiple tumoral lesions in both specimens.The tumor consisted of a myomatous component where the nodules were composed of spindle cells with fascicular array, and a lymphangiomatous component where epithelioid cells could be observed. Immunohistochemically, smooth muscle markers (desmin and SMA) were positive and the epithelioid component showed HMB-45 positivity. A diagnosis of leiomyomatosis-like lymphangioleiomyomatosis was established due to its morphological and immunohistochemical features, the presence of the tumor in multiple foci, and widespread lymphovascular invasion.The patient had a perforation in her bowel twice during the hospital stay and underwent Hartmann operation and ileocecal resection in 2 different surgical operations.After the second operation the patient developed fever and was diagnosed with SARS-CoV-2 infection. No other complication was observed during her stay and the patient's follow-up was unremarkable.Perivascular epithelioid cell tumors are associated with tuberous sclerosis and can rarely appear in the colon. Therefore, lymphangioleiomyomatosis should be in the differential diagnosis in a tuberous sclerosis patient presenting with a colonic tumor. The TSC often gives rise to perivascular epithelioid cell tumors such as renal angiomyolipomas and lymphangioleiomyomatosis (LAM) of the lung. Gastrointestinal involvement, especially LAM formation, is quite rare in TSC and only 2 cases have been previously reported in the literature.,4 In this report, we present a case with leiomyomatosis-like LAM in both the ileocecal and rectosigmoid locations of the gastrointestinal system.Tuberous sclerosis complex (TSC) is an autosomal dominant inherited neurocutaneous syndrome with manifestations in various organ systems due to a loss-of-function mutation in the TSC-1 or TSC-2 genes.2The ileocecal and rectosigmoid resection specimens were fixated in formalin. The specimens were prepared for histopathological examination with the standard paraffin technique and routine hematoxylin & eosin staining. The 3,3′-Diaminobenzidine detection kit was used for immunohistochemical staining. The primary antibodies including clone, dilution, and manufacturer were Desmin , SMA , CD34 , DOG-1 , c-KIT , S100 , ER , PR , and HMB-45 . Convenient positive and negative controls were used. Ethics approval was not required for this report and written informed consent form was obtained from the patient.33.1A 28-year-old woman presented to the emergency department with severe lower abdominal pain that was associated with nausea. Two weeks prior to admission, she had symptoms of abdominal pain and bloody diarrhea which was later diagnosed to be amebiasis. The patient was diagnosed with tuberous sclerosis at the age of 6 months when she developed arrhythmia related to a cardiac rhabdomyoma. The rest of the medical history included adenoma sebaceum, Ash leaf spots, bilateral renal angiomyolipomas, left-sided retinal hamartoma, and allergic rhinitis. She was previously operated 3 times for the removal of multiple renal angiomyolipomas and underwent partial cystectomy due to invasion of the tumor. Physical examination was normal. The laboratory work-up was unremarkable except for anemia (hemoglobin 9.6 g/dL) and an elevated C-reactive protein value of 125 mg/dL. The computerized tomography (CT) scan revealed extralumination of the rectal contrast material at the rectosigmoid region, indicating bowel perforation. The patient underwent proctosigmoidectomy and formation of an end colostomy together with the closure of the anorectal stump (Hartmann operation). Two weeks after the operation, the patient developed a new onset abdominal pain. The CT scan revealed contrast extralumination in the distal ileum and the patient underwent ileocecal resection due to a new onset bowel perforation and severe adhesions. Several days after the second operation she developed fever and was diagnosed with SARS-CoV-2 infection for which she took treatment. Both of the rectosigmoid resection specimen and the ileocecal resection specimen were reported as leiomyomatosis-like lymphangioleiomyomatosis  consisted of full-thickness hemorrhage extending 7 cm proximally from the surgical margin. Macroscopically, there was a 3 cm perforation area in the central part of this hemorrhagic region. Pseudopolypoid lesions ranging from 0.1 to 0.3 cm in diameter were observed in the surrounding mucosa. The ileocecal resection material (ileum 4 cm and colon 16 cm in length) consisted of a 5 cm, pale-colored, nodular lesion in the cecum. Numerous pale nodular lesions, ranging from 0.3 to 5 cm in diameter extending from the mucosa to the subserosa were observed in the cecum. In all resected specimens, the intestinal wall was diffusely thickened and nodular. Infiltration into the subserosal adipose tissue was observed.Histological examination revealed nodular lesions with an extensive growth pattern, extending from the submucosa to the mesorectal adipose tissue in some areas, without affecting the mucosa. Microscopically, the nodules consisted of spindle cell bundles with prominent elongated nuclei organized into branching short fascicles Fig. . Necrosi4 In the gastrointestinal tract, they are most commonly seen in the esophagus and anorectum. They are often differentiated from other spindle cell mesenchymal neoplasms such as gastrointestinal stromal tumor (GIST) by immunophenotyping. GISTs are positive for CD34, CD117, and DOG-1, while leiomyomas are negative with immunohistochemical staining.Leiomyomas are benign stromal tumors characterized by smooth muscle proliferation that are frequently observed in the extremities and arise from the deep subcutis or skeletal muscle. Lymphangioleiomyomatosis is a rare tumor that is caused by the proliferation of perivascular epithelioid cells around lymph nodes and interstitial lymphatics. While lymphangioleiomyomas express localized lesions, lymphatic chain involvement in large segments is referred to as lymphangioleiomyomatosis.Leiomyomatosis refers to the proliferation of smooth muscle cells forming focal tumors at various organ systems throughout the body. Perivascular epithelioid cell tumors (PEComa) are an expanding family of tumors that differ from each other morphologically and immunohistochemically. This tumor family includes angiomyolipoma, lymphangioleiomyomatosis, clear cell sugar tumor of the lung, and a number of spindled and epithelioid neoplasms.–11 In addition, p53 mutations can be seen frequently in TSC-2 mutant malignant PEComas.,12 Infrequently, TSC-2 non-mutant PEComas shelter translocations containing transcription factor binding to IGHM Enhancer 3.PEComas are usually associated with TSC-2 and rarely TSC-1 loss-of-function mutations. These mutations lead to the activation of the mammalian target of rapamycin pathway.,4 Goh et al reported leiomyomatosis-like LAM of the ascending, transverse, and descending colon along with bilateral renal angiomyolipoma in a female patient with tuberous sclerosis complex. Similar to our findings, the smooth muscle cells stained positive for HMB45 and negative for CD34 and CD 117, which suggested leiomyomatosis-like LAM. Kolin et al described a case with tuberous sclerosis complex with a synchronous L-cell type rectal neuroendocrine tumor and leiomyomatosis-like LAM of the rectum. In the report, the immunostaining of the leiomyomatosis-like LAM lesion showed positivity for desmin, caldesmon, HMB-45, and CD117, and showed negativity for CD34.In a patient with tuberous sclerosis, gastrointestinal leiomyomatosis-like LAM is extremely rare and to our knowledge only 2 reports involving the large bowel have been previously reported in the literature.The tumors in our case consisted of both spindle cell proliferation with fascicular array and elongated nuclei and perivascular epithelioid groups. The myomatous component also infiltrated the lymphovascular structures in some areas. Mitotic activity and nuclear pleomorphism were not observed in the cells forming the tumor. In the spindle cell component, smooth muscle markers were positive and GIST markers were negative. HMB-45 expression was observed in the epithelioid component. As a result of these findings, we interpreted our case as leiomyomatosis-like lymphangioleiomyomatosis since the lesions were observed simultaneously in both rectosigmoid and ileocecal resections and lymphatic invasion was present in large segments.–15 Treatment of TSC-related angiomyolipomas should take into account the clinical context and the tumor characteristics and usually include the use of a mammalian target of rapamycin inhibitor together with a nephron-sparing surgery for the conservation of normal renal function. Data regarding the treatment and prognosis of gastrointestinal LAM in TSC patients is lacking but surgery is warranted in the case of an emergency such as bowel perforation observed in this patient.Since LAM formation in tuberous sclerosis commonly occurs within the lung, treatment is usually directed towards improving lung function. Sirolimus has been shown to slow the decline in lung function in patients with LAM, while everolimus is a second-line agent which is used in the treatment of TSC-related angiomyolipomas and central nervous system tumors.4.1Written informed consent was obtained from the patient for publication of the case details and accompanying images.Conceptualization: Ergin Erginoz, Halit Eren Taskin, Gokce Hande Cavus, Abdullah Kagan Zengin.Data curation: Ergin Erginoz, Halit Eren Taskin, Gokce Hande Cavus, Abdullah Kagan Zengin.Formal analysis: Ergin Erginoz, Halit Eren Taskin, Gokce Hande Cavus.Investigation: Ergin Erginoz.Supervision: Gokce Hande Cavus.Visualization: Ergin Erginoz, Gokce Hande Cavus.Writing – original draft: Ergin Erginoz, Halit Eren Taskin.Writing – review & editing: Ergin Erginoz.


Abdominoscrotal hydrocele (ASH) consists of fluid-filled intercommunicating inguinoscrotal and abdominal sac with a characteristic hourglass-like picture on CECT, which usually affects single testis and a rare cause of abdominoscrotal cystic swelling. The precise etiology of ASH is not known. Ultrasonography is the initial diagnostic modality of choice as it demonstrates the intercommunication between the two sacs and also identifies any abnormality of the testis and genitourinary tract. We are reporting a case of a 27-year-old patient presented in the General Surgery OPD of Acharya Vinoba Bhave Hospital in 2019 with bilateral scrotal and abdominal swelling. On examination, cross fluctuation was positive between left hydrocele and abdominal swelling, raising suspicion of ASH, which was confirmed on CECT. The patient underwent excision of sac through left inguinoscrotal approach and an uneventful postoperative course. Abdominoscrotal hydrocele is a rare clinical entity seen only in 0.18%-3.1% of hydrocele cases . ASH is A 27-year-old male patient presented in the General Surgery OPD of Acharya Vinoba Bhave Hospital in 2019 with bilateral scrotal and lower abdominal swelling , which wSince the first description by Dupuytren in 1834 when it was called hydrocele en bisac, many titles have been used until Bickle in 1919 suggested the abdominoscrotal hydrocele as a proper descriptive term . TesticuUSG is the initial modality of investigation in which ASH appears as an anechoic homogeneous lesion and intercommunication can be delineated on graded compression during USG , 6. The ASH should be differentiated from hernia, chord lymphangioma, spermatocele, cystic abdominal mass, and ascites. Long-standing ASH leads to pressure-related complication like hydroureteronephrosis, deep vein thrombosis, leg edema, testicular dysraphism, and impaired spermatogenesis and rarely malignant transformation , 7, 8.Malignancy with ASH seen only in 0.86% of cases, because of low incidence and serial pathological examination of specimens to rule out malignancy, could not provide any evidence that ASH is responsible for malignant transformation .Surgical excision of the sac is the cornerstone of treatment in adult patients because of the unlikelihood of spontaneous resolution, to avoid pressure-related complications . DiffereASH is a rare cause of abdominoscrotal swelling, which has different etiological hypotheses and multiple clinicopathological variants. Clinical examination and USG are initial modalities sufficient to make diagnoses, but CECT is required to prove intercommunication and other associated complexities. Despite available minimal invasive techniques for treatment, complete excision of the sac via inguinoscrotal incision remains the standard approach.


Perioperative high dose rate brachytherapy is a radiotherapy treatment technique which involves intraoperative insertions of brachytherapy catheters into the tumor bed during the surgical resection followed by treatment in the post-operative period. We report here two cases to highlight its use in the primary treatment and reirradiation of head and neck cancers. Brachytherapy using iridium interstitial implants has been practised in head and neck cancers. It has also been combined with surgical resection of tumors with treatments delivered either intraoperatively or perioperatively. Perioperative high dose rate brachytherapy (PHDRB) is a technique which involves intraoperative insertions of brachytherapy catheters in the tumor bed at time of surgical resection followed by a fractionated brachytherapy treatment in the perioperative period.2 It combines the advantages of a highly conformal radiation dose delivery with a clear visualization and demarcation of the tumor bed at the time of surgical dissection. This coupled with incorporation of CT-based treatment planning gives it a high level of treatment delivery precision. Perioperative brachytherapy is an established technique of delivering radiotherapy treatments in sarcomas and its application has been reported in pancreatic cancers.3 Its use in head and neck cancers has been infrequent due to the complex regional anatomy, surrounding vascular and nervous tissues, multitude of organs at risk and the invasive nature of the procedure. PHDRB can be used in head and neck cancers to primarily irradiate the tumor bed to a very high dose of radiation as a single modality or combined with external beam radiotherapy in both the primary treatment and for reirradiation of recurrent cancers.4 We report here two cases highlighting the applications of perioperative brachytherapy in these clinical situations.The first reported application of using a radioisotope to treat a malignancy intraoperatively is credited to Robert Abbe in the year 1910.100 - V150] / V100) of 0.60 was achieved on planning (V100 & V150 represent tissue volume encompassed by 100% and 150% prescription isodose). Brachytherapy treatment was started from the fourth post-operative day on microSelectron HDR  and treatments were delivered twice a day at 6-hourly intervals for a total of 7 fractions of 3 Gy each The first case was a 45 years male who presented with a non-healing ulcer in posterior aspect of the right oral cavity of 2 months duration. The patient was evaluated in the multidisciplinary head and neck cancer tumor board of our institute. Examination revealed a right level Ib lymph node of 2 × 2 cm which was firm mobile, non-tender. Local examination of the oral cavity showed an ulceroinfilitrative growth 6 × 4 cm in size involving the right retromolar trigone region. Biopsy was suggestive of squamous cell carcinoma. Patient was diagnosed as a carcinoma of the right retromolar trigone cT3N1M0 (Stage III). The patient was planned for surgery with post-operative radiotherapy using a combination of perioperative and external beam radiotherapy in view of anticipated close margins. He underwent a wide local excision and right inferior maxillectomy along with right modified neck dissection Type III with right segmental mandibulectomy  . Frozen 2 D1, 22 in the year 2014. The radiation was delivered using conventional radiotherapy with two parallel opposed head and neck treatment portals. He presented with a mass in the right lower neck of 2 month duration in 2017. On examination, there was left level Vb 5 × 5 cm lymph node mass. It was firm and fixed to underlying structures. Contrast-enhanced CT scan was suggestive of infilitration into adjoining soft tissues and biopsy revealed a squamous cell carcinoma. Patient was staged as a recurrent carcinoma base of tongue rT0N2bM0 (rStage IVA). The patient was planned for surgical excision with modified neck dissection and reirridation using perioperative brachytherapy. The indications for perioperative brachtherapy were extracapsular spread of disease with infiltraion into the sternocleidomastoid muscle and an anticipated doubtful R0 resection. The patient underwent modified neck dissection with excision of nodal mass. Five brachytherapy catheters were inserted over tumor bed with intercatheter spacing of 1.5 cm . He had received radical radiotherapy to a dose of 70 Gy over 35 fractions delivered in 7 weeks along with concurrent Inj. Cisplatin 100 mg/ mf 1.5 cm . The pattumor bed effect’ theory explains the rationale of delivering a high dose of radiation in the immediate perioperative period. It alters the interaction of the microscopic residual disease cells within the tumor bed with the normal host tissues and prevents their reimplantation and subsequent local recurrences. Improved local control in turn lead to decreased local and distant failures and improved overall survival.6 Reducing the treatment-related toxicity is another argument for the practice of brachytherapy, as it confines the radiation dose to a small area with a rapid dose fall off in surrounding structures. Concurrent chemoradiation is considered the standard of care in management of locally advanced head and neck cancers. Significant Grade 3 toxicity ranging from 21 to 38% has been reported with the use of concurrent chemoradiotherapy protocols in head and neck cancers. Grade 3 toxicity upto 39% has been reported with reirradiation using external beam techniques.7 One of the ways to reduce this treatment associated toxicity is to reduce the volume of irradiation by integrating brachytherapy into the treatment protocols.The isodose distribution of PHDRB implants 4PHDRB exemplifies the true interdisciplinary management of cancers by integrating brachytherapy with surgery. The ‘8 Reirradiation has been effectively used to manage recurrent head and neck cancers6 and PHDRB is an excellent modality to be used in this indication.Single plane PHDRB implants can deliver a high targeted dose to positive margins and can also increase the surgical margins by 1–1.5 cm thereby improving the local control.9 An added advantage of brachytherapy implants is that they are not affected by organ motion or respiratory movements.Non-nasopharangeal head and neck cancers are a locoregional disease with reported recurrence rates as high as 50% after curative treatment.10 To help patient selection, the University of Navarre predictive model can also be used which divides patients into low risk, intermediate, high and very high risk categories.11 Preplanning with the surgical team is useful and should also review the reconstruction procedure and type of surgical flaps to be used. Identifying the feeding vasculature for the surgical flap can prevent unintended irradiation to the flap vasculature and have implications on flap viability. Tumor location may be the limiting factor for selecting patients in head and neck cancers and PHDRB should be done in a site which allows easy catheter entry and exit without undue bending or knicking of catheters. A vast majority of implants in head and neck will be single plane with an aim to irradiate only the tumor bed as a target area. 9Being an invasive treatment modality, perioperative brachytherapy needs a careful patient selection when being implemented in clinical practice. Patient selection for PHDRB can be aided by dividing patients requiring brachytherapy for reirradiation or those with inadequate surgical margins at difficult resection sites or those with adequate surgical margins as a means of dose escalation.4 Martinez et al reported the 4 year local control rate and overall survival were 85.6 and 46.4%, respectively in head and neck cancers treated with reirradiation using PHDRB.13,14 Treatment-related acute side-effects include bleeding, fistula, graft failure, delayed wound healing. Late morbidity can occur in the form of fibrosis, soft tissue necrosis and osteoradionecrosis. Overall, high grade toxicity has been reported to be between 15 and 69% in single modality procedures and 2.8–30.5% in combined modality procedures.15Periopertive high dose rate brachytherapy alone after R0 resection has been associated with 9 year cancer-specific survival rate of 47.9%.This case report demonstrates the clinical application of PHDRB in head and neck cancers and can be considered as one on the treatment options in patients with high risk margins or where reirradiation is being planned after surgical resection.PHDRB allows brachytherapy catheter placement in anatomical regions not easily accessible to conventional interstitial brachytherapy, since catheters are placed intraoperatively under direct tumor bed visualization.Overall treatment time can be reduced by integration of PHDRB with external beam radiotherapy.It is a versatile treatment to be used for increasing the surgical margins or for dose escalation in primary and recurrent head and neck cancers.


Hypercalcaemia, a condition with abnormally raised calcium levels, is commonly caused by cancer, immobility, certain supplements andother diseases such as sarcoidosis. In this case report, we present a 65-year-old female who presented with hypercalcaemia, hilar adenopathyon chest X-ray and a pathological fracture of her ankle that was unexpectedly due to hyperparathyroidism. A 65-year-old female presented with a fractureof her left ankle following a motor vehiclecollision. She required anticoagulation as shedeveloped a deep-vein thrombosis as a resultof her immobility. She had been complainingof generalised bone pains over the past fewmonths prior to the event, which she hadattributed to old age. As radiological evidenceof significant osteoporosis was found, aconservative management approach wasadopted for the fracture.She was clinically well and showed noevidence of bleeding or skin rashes onexamination. There was swelling aroundher ankle with decreased range of motion.There was no evidence of neurological orvascular compromise. The respiratory systemexamination was normal.2) andserum angiotensin converting enzyme levelof 23 U/L. Her total vitamin D level was atthe lower limit of normal at 73.08 nmol/L(sufficient level >72.5 nmol/L).Her chest X-ray revealed hilar fullnesswith the left being more prominent thanthe right A parathyroid sestamibi scan revealed anarea of hyperplasia within the mediastinum.A computed tomography scan confirmedthe presence of a lesion situated at the lefthilum. No enlarged parathyroid glands wereobserved in the neck. A dual energy X-rayabsorptiometry scan confirmed the presence of osteoporosis with a t-score of –4.3 in thespine and –4.8 in the hipShe received a dose of zoledronic acidfor the osteoporosis and saline diuresis pre-operatively to decrease the serum levelsof calcium. She underwent video-assistedthoracoscopic surgery (VATS) to remove themediastinal mass. Following this, the serumcalcium levels decreased to 2.19 mmol/Land the PTH decreased to 15.6 pmol/L. Thehistology confirmed the presence of ectopicparathyroid tissue that had atypical cells withclear and eosinophilic cytoplasm and stippledchromatin with the round and uniformnuclei. The atypical endocrine cells stainedpositive for chromogranin, synaptophysinand parathyroid hormone.Unfortunately, she developed a post-operative complication – on re-initiation oftherapeutic anticoagulation, she bled fromthe vessel supplying the mass. She developeda haemothorax which required drainage aswell as a blood transfusion but made a fullrecovery thereafter.[1]The differential diagnosis of a hilar massrequires exclusion of possible causes such asinfectious pathogens, cancer (carcinomas orlymphomas) and sarcoidosis. The coexistenceof a hilar mass with hypercalcaemia maysuggest that the latter two are the more likelycauses.[2]Hyperparathyroidism is an underdiagnosedcondition in the developing world. In theSouth African setting, the median age is 60years with a female predominance of 78.6%.Most of the patients are symptomatic andsurgery remains the mainstay of therapy.[3] and alwayspose a treatment and diagnostic challenge.A combination of radiological investigationshave been utilised to increase the sensitivityand specificity of the diagnosis and facilitatethe localisation of these lesions prior tosurgical removal. Accurate localisation hasimproved the success of minimally invasivesurgery (VATS) and has reduced the need for median sternotomy to reach these hard-to-locate lesions.[4]Althougha diagnosis might be suspected, histological confirmation is requiredto make a correct diagnosisEctopic lesions occur in up to 20% of allcases of parathyroid adenoma[5]The majority of ectopic glands are found in the mediastinum, whichis related to its embryonic development. When ectopic tissue descendsinto the mediastinum, it does so with the thymus during the fifth weekof development. It is usually located below the level of the clavicle,with the deeper ones posing more of a challenge. The inferior glandsare located in the anterior mediastinum, while the rarer superiorglands are found in the posterosuperior mediastinum.In this case, the cause for hypercalcaemia was unexpected and notone usually included in the differential possibilities of a respiratoryphysician.


Primary central nervous system (CNS) tumors represent the most common solid tumors in childhood. Ependymomas arise from ependymal cells lining the wall of ventricles or central canal of spinal cord and their occurrence outside the CNS is extremely rare, published in the literature as case reports or small case series. We present two cases of extra-CNS myxopapillary ependymomas treated at our institution in the past three years; both cases originate in the sacrococcygeal region and were initially misdiagnosed as epidermoid cyst and germ cell tumor, respectively. The first case, which arose in a 9-year-old girl, was treated with a surgical excision in two stages, due to the non-radical manner of the first operation; no recurrence was observed after two years of follow-up. The other case was a 12-year-old boy who was treated with a complete resection and showed no evidence of recurrence at one-year follow-up. In this paper, we report our experience in treating an extremely rare disease that lacks a standardized approach to diagnosis, treatment and follow-up; in addition, we perform a literature review of the past 35 years. ZFTA fusion-positive; supratentorial ependymoma, YAP1 fusion-positive; posterior fossa ependymoma; posterior fossa ependymoma, group PFA; posterior fossa ependymoma, group PFB; spinal ependymoma; spinal ependymoma, MYCN-amplified; myxopapillary ependymoma; subependymoma. In particular, myxopapillary ependymoma is now considered CNS WHO grade 2 instead of 1, since its likelihood of recurrence is currently known to be similar to that of conventional spinal ependymoma.Ependymoma comprises approximately 9% of all pediatric central nervous system (CNS) tumors, representing the most common spinal cord neoplasm of this age group . It origAmong all pediatric brain and spinal tumors, ependymoma occasionally arises outside the CNS. Various localizations are described in the literature: the most common during childhood is the subcutaneous tissue of the sacrococcygeal region, whereas less frequently is the presacral region; rarer localizations can include the mediastinum, liver, or lung, described mainly in adulthood ,4,5. HerA 9-year-old girl presented at an outside institution with a history of a persistent mass in the gluteal region; no associated symptoms were reported. The presence of a congenital sacral dimple was reported in the anamnesis. She underwent lumbosacral magnetic resonance (MRI) which revealed the presence of T2 hyperintense capsular formation measuring 30 × 15 mm in the soft tissues of the left paramedian posterior sacrococcygeal region, without any intrinsic spinal cord component nor connection to the central canal. An epidermoid cyst was hypothesized, and the girl was then subjected to surgical removal of the lesion. Surprisingly, histological examination revealed a moderately cellular neoplasm with areas of fibromyxoid stroma. An immunohistochemical analysis showed the neoplastic cells were strongly positive for glial fibrillary acidic protein (GFAP) and S-100, whereas they were negative for cytokeratin (AE1/AE3), epithelial membrane antigen (EMA) and Olig2. These features allowed for the diagnosis of myxopapillary ependymoma. Proliferation index evaluated with Ki-67 was 2–3%, reaching values of 9–10% in the central part of the neoplasm. The surgical margins were positive, showing the non-radicality of the resection. Due to the rarity of the neoplasm, the girl was then referred to our center. Because myxopapillary ependymoma has the potential for metastatic spread [A 13-year-old boy with a history of a congenital slow-growing sacrococcygeal swelling, sought care for an increase in volume and one episode of self-limiting inflammation of the lesion in the last two months A. UltrasEpendymoma represents the third most frequent CNS tumor in childhood and is mainly localized in the posterior cranial fossa where spinal localization is more frequent among adults ,8. The bWhile it is universally accepted that CNS ependymomas develop from the ependymal cells lining in the ventricles and the central canal, the pathogenesis of extraspinal ependymomas is still debated, with three main hypotheses reported in the literature ,4,6 Fig. The firThe clinical presentation varies according to the onset region: in their review of sacrococcygeal ependymomas, Lien and colleagues found an asymptomatic mass as the most frequent initial presentation (about 50% of cases), while rarer symptoms such as pain, tenesmus, constipation, or signs of neuropathy were encountered ,34,44. TCurrently, there are no standardized guidelines for the management of extra CNS ependymomas. The most effective treatment seems to be complete surgical resection: Sonneland et al. in their study on 77 cases found a lower relapse rate in patients undergoing en bloc resection than in those undergoing piecemeal resection (10% vs 19%). Gross total resection is associated with better OS compared to subtotal resection (19 years vs. 14 years) . CoccygeThe role of radiotherapy in the treatment of pediatric extra CNS ependymomas is less clear than that of surgery. Unlike its demonstrated role in the treatment of the intracranial disease, there is no clear evidence regarding its real effectiveness in the extra CNS setting. However, there is agreement in the literature to recommend it in the case of subtotal resection or inoperable mass, after the removal of recurrent lesions and in the case of metastatic disease ,5,21,48.The role of chemotherapy in treating children affected by intracranial ependymomas remains unproven despite intensive investigation ,53. ThusDespite the generally good prognosis of this disease, a long-term follow-up must be warranted, given the aforementioned possibility of relapse even after many years.Extra-CNS ependymoma is exceedingly rare and difficult to diagnose by imaging. The rate of metastastic disease is not trivial and warrants total body stadiation in all cases. The mainstay of treatment is radical surgery, including resection of coccygeal bone when involved by infiltrative margins. The role of adjuvant treatment is limited to selected cases. Collaborative studies are needed to better define the optimal management of these patients.


Patients commonly present with neck masses to the Emergency Department. Theacute presentation of such a mass can be alarming to patients and theirfamilies. In this report we discuss a rare etiology of an acutely presentingneck mass in an adult.We present a 19-year-old patient with an acute neck mass. The mass developedabruptly soon after initiation of a new upper body strength-trainingregimen. The patient’s history was unremarkable for any trauma orconstitutional symptoms. Physical examination revealed the mass, which wasdiagnosed as a lymphatic malformation by imaging. Surgical removal wassuccessful with pathology confirming the diagnosis.Lymphatic malformations, although rare, may present in adulthood. The acutepresentation of a new mass, coupled with a lack of concerning constitutionalsymptoms, should increase the diagnostic suspicion of a lymphaticmalformation. Neck masses are a common presenting complaint among adult patients.An otherwise healthy 19-year-old female presented to an outlying emergency department(ED) on the same day that she noted an acutely developed mass on the distal rightneck . ThepatThe patient presented to our ED the following day as concern grew for the lack ofdefinitive diagnosis, and she was evaluated again. Once again, she denied trauma,constitutional symptoms, or difficulty swallowing. She noted the mass was new andfirst discovered while showering the day prior. She had recently initiated a newstrength-training regimen for her upcoming athletic season, which focused on upperbody development 14 days prior to discovery of the neck mass. She was afebrile at36.8° Celsius with blood pressure of 131/54 millimeters of mercury, pulse of64 beats per minute, respiratory rate of 14 breaths a minute, and oxygen saturationof 100% on room air.Physical examination revealed a large, easily palpable, nonerythematous, slightlytender mass that extended from eight centimeters (cm) proximal to the clavicle tothe supraclavicular region. There was no palpable associated lymphadenopathy.Laboratory studies included complete blood count with white blood count of 10thousand cells per cubic millimeter (K/uL) (reference range: 3.4–9.4 k/uL),platelets of 175 K/uL (140–410 K/uL), hemoglobin of 13.5 grams per deciliter(gm/dL) (12–16 gm/dL), erythrocyte sedimentation rate of 4 millimeters perhour (mm/hr) (0–20 mm/hr); thyroid-stimulation hormone of 1.15microinternational units per milliliter (μIU/ML) (0.45–5.33μIU/ML); and mononucleosis test negative.Computed tomography (CT) of the neck and soft tissue revealed a large cysticcollection along the right neck deep to the sternocleidomastoid muscle and extendinginferiorly into the supraclavicular region with two smaller, adjacent cysticcollections. No aerodigestive tract mass or cervical lymphadenopathy was noted. Theprimary consideration was a large lymphatic malformation. Outpatient otolaryngologyfollow-up was arranged after they reviewed the CT results and concurred with thepresumed lymphatic malformation diagnosis.What do we already know about this clinical entity?Lymphatic malformations are usually diagnosed within the first two yearsof life.What makes this presentation of disease reportable?This was an acute presentation of a lymphatic malformation in an adultwhich is unusual in both acuteness and age of the patient.What is the major learning point?Lymphatic malformation should be considered in the differential diagnosisof an acutely presenting neck mass regardless of age.How might this improve emergency medicine practice?Emergency physicians can provide reassurance that acutely presentingmasses lacking constitutional symptoms, recent illness, or associatedlymphadenopathy may portend a better prognosis.The patient subsequently underwent magnetic resonance imaging (MRI) that wasconsistent with a lymphatic malformation . ApproxiAfter recovering from the surgery, the patient began physical therapy for hershoulder. At 10 weeks post-surgery, the patient was continuing with physical therapyand able to return to her collegiate crew participation.The differential for neck masses can be differentiated by acuity of presentation.Acute masses are most often infectious or traumatic. Subacute or chronic masses aremore often considered to be malignant or related to other chronic illness .1111Lymphatic malformations are uncommon, benign masses that result from abnormallymphatic system development. The preferred term lymphatic malformation encompassescystic hygromas, lymphangiomas, cavernous lymphangiomas, cystic lymphangiomas, andlymphangioma circumscriptum. In the majority of cases, these masses are recognizedby two years of age, where prevalence is believed to be approximately 1:4000 livebirths.Diagnosis of lymphatic malformations usually requires advanced imaging. Determiningwhich imaging modality to choose can be difficult for the emergency physician.Ultrasound can determine cystic structures but lacks the ability to reliablyestablish mass etiology.Treatment options for lymphatic malformations include percutaneous drainage, surgery,sclerotherapy, laser therapy, and radiofrequency ablation.9We report a case of a lymphatic malformation that presented acutely as a new neckmass in an adult. Rapidly developed new-onset masses should be evaluated as possiblelymphatic malformations. The lack of constitutional symptoms and rapid onset makemalignant neoplasm less likely. Advanced CT and MRI imaging led to the diagnosis andhelped to prepare for successful surgical excision.


The anterolateral abdominal Hernias are a frequent reason for consultation; Spiegel's hernia is a rare spontaneous abdominal anterolateral hernia  for patients between 40 and 70 years old, There are risk factors such as intra-abdominal hyperpressure secondary to morbid obesity, multiple pregnancies and chronic cough. The surgery is the standard treatment; whether by raphy or prosthetic mesch. We report the case of a 42 year old male admitted to the emergency room for an occlusion syndrome due to the strangulated spiegel hernia with caecal and appendicular contents. ∙Hernias of the anterolateral abdominal wall are a frequent reason for consultation;∙Spiegel's hernia is a rare form of spontaneous abdominal anterolateral hernia  in subjects between 40 and 70 years of age∙There are predisposing factors such as intra-abdominal hyperpressure secondary to morbid obesity, multiple pregnancies and chronic cough, the treatment is surgical; by raphy or by prosthetic plasty. The physical examination found an apyretic patient, distended abdomen with the presence of a painfull mass of 8 × 5cm in the right iliac fossa, irreducible. The pelvic digital examination revealed an empty rectal ampulla. Biology assessments found a hyperleukocytosis with 12550 white blood cells/ml. The diagnosis of a strangulated spiegel hernia was retained. Surgical exploration, by local incision, found an 8cm hernial sac with a 2cm hole at the junction of the lateral edge of the rectus maximus and the transverse muscles of the abdomen with the caecum and appendix herniation. An appendectomy was performed and the caecum moved back in the abdominal cavity after resuscitation. The peritoneal sac was resected. Closure of the orifice by a 2 resorbable interrupted suture was performed Fig. 1perClosure of the aponeurosis of the external oblique muscle with interrupted stitches with absorbable thread. A subcutaneous redon drain was installed and the postoperative was simple. The patient was discharged on the third postoperative day after transit retake and drain removal. the patient was seen after one month in a good health.2trans-abdominal-preperitoneal or extra-peritoneal raphy or mesch repair. If the hernial ring is narrow (less than 2 cm) A direct approach herniorrhaphy is usually sufficient. Otherwise, a laparotomy approach or transperitoneal laparoscopic repair with a double-sided or pre-peritoneal mesch are performed, with a lower risk of recurrence with synthetic mesch than simple suture [Spiegel's hernias are rare and correspond to the protrusion of a peritoneal sac through an acquired or congenital anatomical orifice of the Spiegel's line located at the crossing of the fibres of the transverse and oblique abdominal muscles on the lateral edge of the rectus abdominis . This wee suture .3Spiegel hernia is a rare pathology. Strangulation remains a rare but serious complication that can be life-threatening. Abdominal CT scan allows the diagnosis and reveals complications, in particular of strangulated hernia.As per international standard written ethical approval has been collected and preserved by the author(s).None.This work was carried out in collaboration among all authors. All authors contributed to the conduct of this work. They also declare that they have read and approved the final version of the manuscript.DR EL ATTAR LAYLA.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.Researchregistry2464.Authors have declared that no competing interests exist.


Pulmonary adenoid cystic carcinoma (PACC) is an exceedingly rare tumor of low-grade malignancy. Diagnosis is often late, so the tumoral process may be huge at presentation. Surgical resection could be challenging, and the recurrence rate would be greater. We report, hereby, a case of proximal PACC with involvement of the carina in a young male adult, without respiratory distress. Surgical resection was performed through a left pneumonectomy followed by a complex trachea-bronchoplastic procedure. During the operative time, the assisted ventilatory mode was carefully chosen. No adjuvant treatment was needed. Our patient is still under clinicoradiological surveillance and remains disease-free. Pulmonary adenoid cystic carcinoma (PACC) is an exceedingly rare tumor of low-grade malignancy, accounting for 0.09 to 0.2% of all lung cancers . DiagnosA 29-year-old male patient with no past medical history was admitted to our department to explore a right basal thoracic pain mentioned two months ago aggravated by an acute onset dyspnea. He was an active smoker and worked as a baker. On examination, the patient was afebrile and eupneic, no oxygen was needed, and auscultation found a decrease in vesicular murmur on the left with dullness on percussion. Chest X-ray revealed a homogeneous pulmonary opacity occupying the entire left pulmonary field with a mediastinum lifted to the left. Bronchial endoscopy revealed a hypervascularized bud obstructing the entrance to the left mainstem bronchus . The anaThe patient underwent a left pneumonectomy with a section of the left mainstem bronchus and lymph node dissection through a left thoracotomy. Then, a right thoracotomy was performed. Selective high-frequency jet ventilation was performed through the right main bronchus. The trachea-bronchial section was enlarged to the lower part (4 cm) of the trachea and the carina. A terminoterminal anastomosis restored continuity of the distal part of the trachea and the right mainstem bronchus. Anastomotic sutures using 4-0 polydioxanone suturing thread were placed around the entire circumference, then ligated making ends meet.Postoperatively, the patient was given oxygen therapy at home for a month. The CT scan performed 9 months after surgery showed no signs of regional or distant recurrence. Endoscopic control objectified a barely visible bulge at the right strain bronchus that marks trachea-bronchial plasty without narrowing of the lumen. A clinicoradiological follow-up is ensured for 24 months nowadays . The patPACC is uncommon but represents the predominant type of salivary gland-type lung carcinomas . So far,ACC develops mostly in the trachea, the mainstem, or the lobar bronchi , 7. The Histologically, ACC shows differentiation towards ductal and myoepithelial cells . AuthorsACC tends to submucosal extension linked to the arrangement of the accessory salivary glands, located between the cartilage and the tracheal fibromuscular membrane, at the level of the posterior wall of the trachea. It shows up in CT scan as an intraluminal mass extending through the tracheal wall. Sometimes, the lesion may present as a circumferential parietal thickening , 9. RespTreatment options for advanced PACC patients are limited. Currently, the best treatment relies on complete resection with tracheobronchial plasty depending on tumoral location and extension. Developed surgical and anesthesiological procedures would make even wide tumors resectable. Contraindications to surgery include mediastinal lymph nodes, vascular involvement, tracheal invasion for more than 50% of its length, and distant metastases .The extent of surgical resection and adjuvant radiotherapy may influence the prognosis of ACC. In a 30-year study of ACC of the airway, Calzada et al. found a high progression-free survival rate for patients who underwent surgical resection followed by adjuvant radiotherapy . Early rLung adenoid cystic carcinoma is a rare lung cancer with protracted but unpredictable growth behavior. Diagnosis and treatment are not yet fully consensual. For huge resectable masses, postoperative radiation would cure the microscopically residual tumor of the mediastinum. The mainstay of treatment is based upon combined surgical resection to radiotherapy. A long-term follow-up is required due to the risk of local or distant recurrence.


We report a case of a large, ulcerating proliferating trichilemmal cyst in a 63-year-old woman, with clinical, radiological, macroscopic, and microscopic correlation. The outbreak of the Coronavirus pandemic delayed her treatment. We review the literature on proliferating trichilemmal cysts, which are relatively rare tumors, which generally are considered benign. However, we found a high rate of malign cases, which stresses the importance of rapid surgical excision and histological diagnosis. Even though our proband had delayed treatment, the tumor did not transform to a malignant form. Proliferating trichilemmal cysts (PTCs) are relatively rare tumors that may appear all over the body but most frequently on the scalp in middle-aged women . PTCs ocWe here report the case of a 63-year-old woman with a large PTC, where surgical treatment was delayed due to the outbreak of the Coronavirus pandemic and a review of the relevant literature on PTCs.The proband is a 63-year-old woman, with ASA 3 status, who rarely left her home due to sequelae from previous apoplexy. She had some minor tumors on the scalp for many years, for which she had not seen a doctor because they did not bother her. When one of them grew within a few months, she went to her General Practitioner (GP) for treatment. Her GP referred her, to the Department of Otorhinolaryngology and Maxillofacial Surgery at Zealand University Hospital (ZUH), Koege, in October 2019, for a lipoma on the scalp measuring approximately 5 cm in diameter (for timeline see Ultrasound in October 2019 showed a slightly inhomogeneous subcutaneous process and fine needle aspiration cytology (FNAC) with blood cells. In June 2020, the ultrasound at a private hospital found a vascularized tumor, and a needle puncture gave plenty of blood. A CT scan from July 2020 showed a marked progression of an extracranial tumor compared to a CT cerebrum, which was performed because the proband had cerebral apoplexy in 2012 . The proBecause of the probands ASA 3 status, she was referred back to the ZUH, Koege, for surgery. She had trouble sleeping due to the anatomy of the tumor and fear of bleeding from the tumor. The proband underwent complete surgical resection of all three lesions. We reconstructed the extensive scalp defect caused by the tumor resection with local fasciomusculocutaneous flaps .Macroscopic examination  showed aAt postoperative control, small central necrosis was present. Due to the long distance to the hospital, the proband wished further wound care to be at her own GP. The wound became infected with Staphylococcus Aureus and was treated with antibiotics. The wound was almost healed by March 2021, where only a minor defect was present as seen in The proliferating trichilemmal cyst was first described as a proliferating epidermoid cyst by Wilson Jones in 1966 . Just moThe common pilar cyst (or TC) occurs in 5–10% of the population, of which 2% become PTCs . PTC is The proliferating trichilemmal cyst appears clinically as a subcutaneous nodule. It can resemble a cyst or lipoma, and the skin can be atrophic or, as in our case, flaking and ulcerated. PTC is often confused with squamous cell carcinoma (SCC). Other differential diagnoses are epidermoid cyst, keratoacanthoma, sweat gland tumor, basal cell carcinoma, angiosarcoma, pilomatrixoma, dermatofibrosarcoma protuberans, and cylindroma . TCs oftFine needle aspiration cytology helps the surgeon differentiate between malignant and benign lesions. Definitive diagnosis can be challenging on FNAC from a cystic lesion, as enough cells are required, and FNAC from PTC is, therefore, often misdiagnosed. This is supported by the cytological study by Shet et al., who found TCs to yield more keratinous debris and sparse to no epithelia on FNAC . TCs shoThe proliferating trichilemmal cyst is composed of variable-sized lobules, which macroscopically give the PTC a honeycomb appearance, with small cysts filled with keratin material . MicroscImmunohistochemistry has been examined widely in different cystic tumors, and PTCs have been found positive for keratin markers K10 and K17 . ImmunohProliferating trichilemmal cysts have been studied with MRI, ultrasound, and CT , 27, 28.Adequate treatment of proliferating trichilemmal cysts includes surgical excision and skilled histopathological examination of the excised tumor for proper diagnosis. When malignancy is present, a 1-cm margin is recommended . Mohs suTypically, TCs are present for years as “simple cysts” before they enlarge over months to years and yield disfiguring tumors that might ulcerate. According to Brownstein , trauma A proliferating trichilemmal cyst is a cyst derived from the more common TC and should be considered when rapid growth occurs. Histological diagnosis might not correlate with the clinical course, and surgical excision should not be delayed because of the risk of malignant transformation. Even when benign, a larger tumor will only complicate surgical reconstruction of the area. The true incidence of malignant transformation of PTC is currently unknown but thought to be very rare . Based oThe original contributions presented in the study are included in the article/supplementary material, further inquiries can be directed to the corresponding author/s.Written informed consent was obtained from the individual(s) for the publication of any potentially identifiable images or data included in this article.CK: general script, literature search, gathering information, and production of figures. PH: specialist consulting, rewriting and revisions, consent from the patient, and help with submission. All authors contributed to the article and approved the submitted version.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.


Osteosarcoma of the jaw only represents 0.5-1% of tumors of the facial mass. Due to its rarity, clinical diagnosis is thus difficult. The guidelines for this pathology remain unclear, and the need for neoadjuvant chemotherapy is still debated. This case report aims to describe a rare case of chondroblastic osteosarcoma in a 50-year-old woman on risedronate treated by neoadjuvant chemotherapy. The tumor extended from the mandibular left first premolar to the mandibular right canine. An excisional biopsy was performed, leading to a diagnosis of chondroblastic osteosarcoma. Neoadjuvant chemotherapy was ineffective, as it did not result in the shrinkage of the tumor. A pelvi-mandibulectomy with fibula free flap reconstruction of the mandible was subsequently successfully performed followed by radiotherapy. Osteosarcoma is one of the most frequent malignant bone tumors ,2. HowevA 50-year-old woman was referred by her dentist for the evaluation of mandibular swelling. The patient had osteoporosis treated with risedronate. Physical examination revealed swelling of the mandibular symphysis with chin deformation. The tumor did not seem to adhere to the skin but extended from the mandibular left first premolar to the mandibular right canine. The anterior buccal floor was deformed, and the inferior alveolar nerve was preserved. An excisional biopsy of the buccal mass was performed, confirming an initial diagnosis of chondrosarcoma. The histological plates were reinterpreted, leading to a final diagnosis of chondroblastic osteosarcoma. Cartilaginous tissue with chondroid matrix was observed in which chondrocytes with cytonuclear abnormalities were found Figure . ImmunohIt was decided to implement neoadjuvant chemotherapy with doxorubicin 80 mg and cisplatin 140 mg over three cycles. Due to the lack of significant evolution in the tumor mass, an interrupted pelvi-mandibulectomy with bilateral lymph node dissection was performed along with reconstruction with a fibula free flap. The histological analysis after the tumor resection showed a lobulated architecture of the tumor, with pleomorphic cells associated with a predominantly cartilaginous matrix and a matrix in the form of thick immature bone areas  repair mechanism , drug inThis article reported a rare case of chondroblastic osteosarcoma in the mandible treated by neoadjuvant chemotherapy. The diagnosis of OSJ remains difficult from both a clinical and histopathological point of view. There are no guidelines regarding the use of chemotherapy and particularly the use of neoadjuvant chemotherapy which was unsuccessful in this case. This type of tumor can, nevertheless, be fatal. Clear standardized recommendations should, therefore, be put in place for the care of these patients.