The junction of the bones of the orbit, caudal maxilla and zygoma intersect to form an anatomically intricate region known as the orbitozygomaticomaxillary complex (OZMC). Given the critical role of the OZMC in the structure, function and esthetics of the skull and midface, tumors in this region present unique challenges to the oromaxillofacial surgeon. Attempts to achieve histologically clean tumor margins in a cosmetically pleasing manner requires excellent intra-operative visualization. Additionally, minimized intra-operative and post-opertive complications is of paramount importance. In this manuscript we describe a combined intra- and extraoral approach to extensive tumors of the OZMC that incorporates orbital exenteration as a technique, which allows for excellent intra-operative visualization and mitigate intra- and post-operative complications. In addition, we describe our experience utilizing the technique in five clinical cases. It is generally accepted that the primary goal of any tumor excision is to obtain tumor-free margins. Maintaining proper function, managing local pain, and achieving good to excellent cosmesis are additional goals. The degree to which one may achieve these goals is dependent on location and biological behavior of the tumor as well as the complexity of the resultant reconstruction procedure.The orbitozygomaticomaxillary complex (OZMC) is the region of the skull where the zygomatic, and maxilla bones as well as bones that contribute to the medial orbital wall  unite . The OZMExcision of OZMC tumors often involves the medial orbital wall, which requires excellent visualization to prevent inadvertent penetration of the rostral cranial fossa. Additionally, significant ophthalmic side-effects, such as enophthalmos, diplopia and entropion, are expected if orbital reconstruction is not pursued –5. The dPrior to surgical planning all patients received a head computed tomography (CT) study with and without contrast , mandibular lymph node aspiration as well as aspiration of any parotid or medial retropharyngeal nodes that showed deviation from normal size or echogenicity. The airway was secured by standard orotracheal intubation immediately following induction of general anesthesia. Due to the nature of an academic anesthesia and pain management service, anesthetic protocols were not standardized and were determined by the attending anesthesiologist. Where indicated, extraction of occluding mandibular teeth was preformed. In order to minimize oral bacterial load and contamination, an oral antiseptic (0.12% chlorhexidine) rinse was applied and the dentition in the region of the surgery were scaled and polished. Intra-operative regional anesthesia using 0.5% bupivicaine was provided with an ultrasound-guided trigeminal nerve block as described by Viscasillas et al.  Dogs werAn extraoral incision was begun ~3 cm rostral to the medial canthus of the eye along the dorsolateral muzzle and extending caudally toward the medial canthus of the eye. The incision was continued caudally, incorporating a transpalpebral incision, and extending along the dorsal aspect of the zygoma . ExenterThe intraoral approach was commenced by incising the gingiva, vestibular and hard palatal mucosa along the intraoral surgical markings. A combination of sharp and blunt dissection was used to incise the submucosal tissue and muscles along the maxilla and zygoma to approach the underlying OZMC. The levator nasolabialis, orbicularis oris, rector anguli oculi, zygomaticus, sphincter coli pars intermedia and frontalis muscles were incised followed by the levator labii maxillaris and caninus, masseter and temporalis muscles. Vessels crossing the surgical margins were double ligated with a 4-0 synthetic monofilament absorbable suture  and transected. A periosteal elevator was used to reflect the periosteum and associated soft tissues off the underlying bones. By joining the intraoral approach with the extraoral approach, this step, in effect, created a composite bipedicle flap .en bloc excision, which included the suborbital tissues and the zygomatic salivary gland when indicated  was infused into the muscular and subcutaneous tissues surrounding the excision to provide post-operative analgesia. The remaining bipedicle flap of the cheek/superior labia was then bluntly dissected to separate the buccal/labial mucosa from the dermis and muscles. An advancement flap was utilized to suture the buccal/labial mucosa to the palatal mucoperiosteum with 4-0 synthetic monofilament absorbable suture  in a simple interrupted pattern  of the head revealed a lobulated, soft tissue attenuating, non-uniformly enhancing 1.6 cm diameter mass with numerous areas of patchy, coalescing mineralization along the palatal aspect of maxillary fourth premolar and molars. The tumor primarily involved the caudal maxilla and palatine bones; advancing dorsally into the pterygopalatine fossa. Previously perform histopathology revealed a diagnosis of osteosarcoma.The case was referred to the Dentistry and Oromaxillofacial Surgery service for surgical excision. The tumor was excised as an en bloc excision of the OZMC and associated musculature, oral mucosa, gingiva and dentition utilizing a combined intra- and extraoral approach with a transpalpebral exenteration. The osseous excision extended from the mesial aspect of the right maxillary third premolar tooth to the mid-zygoma. The medial extent of the excision was the median palatine raphe and the inferior aspect of the medial orbit. Following excision, the wound was closed as described above.Histological evaluation of the resected tissue confirmed the diagnosis of osteosarcoma and surgical margins were free of tumor with a narrow margin (16 mm) at the distal aspect of the excision. The dog recovered uneventfully from general anesthesia despite hypotension that responded to a transfusion of packed red blood cells. The patient remained on a continuous rate infusion (CRI) of fentanyl (3 μg/kg/hr), and ketamine (3 μg/min/hr) overnight. Transdermal fentanyl patches (100 mcg/hr + 25 mcg/hr) and ampicillin/sulbactam (20 mg/kg IV q 8 h) was also administered. The dog was discharged the following day with oral carprofen (2 mg/kg q 12 h), tramadol (5 mg/kg q 8–12 h) and amoxicillin/clavulanic acid (13.75 mg/kg q 12 h). Instructions to feed only canned or softened kibble and to avoid toys or mouth play were given to the owners.At the time of recheck and suture removal 14 days later the owners reported the dog was eating and drinking well. The skin and oral incisions had healed well and no discharge, redness, swelling or dehiscence was noted. The owners elected to pursue definitive radiation therapy over 20 fractions of 2.7Gy to a total of 54Gy. This intensity-modulated radiation therapy was delivered using 6 MV helical tomotherapy  and planned with inverse planning software (TomoTherapy HiArt® v3-5)]. Following radiation therapy, vinorelbine chemotherapy was initiated. Pulmonary metastasis was identified 21 months after the surgical excision, radiation therapy and chemotherapy was initiated. Two weeks later, the patient was euthanized due to poor quality of life secondary to pulmonary disease.A 7.5-year-old neutered male Standard Poodle was referred to the Dentistry and Oromaxillofacial Surgery service for evaluation and biopsy of an oral mass affecting the right caudal maxilla. An approximately 3.5 cm diameter, firm mass was present on the palatal aspect of the right maxillary fourth premolar tooth and first molar tooth. Thoracic CT, mandibular and medial retropharyngeal lymph node aspirations revealed no evidence of metastasis. Head CT revealed a large lobulated, heterogenous, mineral attenuating 5.1 × 3.8 × 2.9 cm mass centered at the right maxillary fourth premolar tooth and first molar tooth. The mass was invading and destroying the palatine, zygomatic and frontal bone as well as the rostroventral ethmoid turbinates, maxillary recess, infraorbital canal and nasopharyngeal meatus . HistopaThe tumor was excised as an en bloc excision of the OZMC and associated musculature, oral mucosa, gingiva and dentition utilizing a combined intra- and extraoral approach with a transpalpebral exenteration. The osseous excision extended from the mesial aspect of the right maxillary third premolar tooth to the mid-zygoma. The medial extent was 7 mm toward the contralateral side from the median palatine raphe and included portions of the maxilla, lacrimal, palatine, pterygoid and frontal bones. Following excision, the wound was closed as described above.Histological evaluation of the resected tissue confirmed the diagnosis of multilobular tumor of bone and tumor-free margins. All margins were reported as clean with at least 1 cm tumor-free tissue, except for the palatal margin, which was reported as narrow (3 mm). The dog recovered uneventfully from general anesthesia with minimal blood loss and was managed overnight on a CRI of sufentanil (0.3 μg/kg/hr), lidocaine (10 μg/min/hr) and ketamine (3 μg/min/hr). Additionally, a subcutaneous dose of carprofen (4.4 mg/kg) was administered. The dog was discharged the following day with a 150 mcg transdermal fentanyl patch, oral tramadol (4 mg/kg q 8-12 h) and carprofen (2.2 mg/kg q 12 h). The owners were instructed to feed only canned or softened kibble and to avoid toys or mouth play.At seven days post-operatively there was significant halitosis noted by owners and evidence of a 3 cm x 12 cm dehiscence at the caudal aspect of the intraoral closure site. The remaining closure sites were healing well and a good cosmetic appearance was apparent. An island axial pattern flap based on the contralateral major palatine artery was used to close the caudal dehiscence and healing was uncomplicated. At 4 months post-operatively recurrence of the neoplasia was noted on CT and owners elected against further surgical intervention. The patient was palliatively managed with the referring veterinarian and euthanized 11 months after the original surgery date.An 8.5-year-old neutered male Siberian husky was referred to the Dentistry and Oromaxillofacial Surgery service for treatment of a left maxillary papillary squamous cell carcinoma. On presentation, a 0.5 cm diameter, round and firm mass was visible in the buccal gingiva of the left maxillary first molar tooth. The patient was placed under general anesthesia for thoracic CT and regional lymph node aspiration, which revealed reactive lymphoid tissue and no evidence of pulmonary metastasis. A head CT was performed and revealed a 1.9 × 3.0 × 2.6 cm soft tissue attenuating, inhomogeneous contrast enhancing nodular mass centered at the left maxillary first molar tooth.The tumor was resected as an en bloc excision of the OZMC and associated musculature, oral mucosa, gingiva and dentition utilizing a combined intra- and extraoral approach with a transpalpebral exenteration. The excision extended from the mesial aspect of the left maxillary fourth premolar tooth distally to the mid-zygoma. The palatal extent of the excision was the median palatine raphe. The excision extended from the inferior orbital crest of the lacrimal bone and maxilla at its dorsomedial extent to the dentition at its ventrolateral extent including the maxilla and dorsal zygoma. Following excision, the wound was closed as described above.Histological evaluation of the resected tissue confirmed the diagnosis of papillary squamous cell carcinoma and surgical margins were free of tumor with a narrow margin (4 mm) at the distal aspect of the excision. While this patient received a whole blood transfusion due to hypotension in the face of only moderate blood loss, the dog recovered uneventfully from general anesthesia. The patient was managed overnight on a CRI of fentanyl (3 μg/kg/hr) and ketamine (3 μg/min/hr). Carprofen (2.2 mg/kg SQ) and cefazolin (25 mg/kg IV q 8 h) was also administered. The dog was discharged the following day with a 75 mcg transdermal fentanyl patch and oral carprofen (2.2 mg/kg q 12 h), tramadol (5 mg/kg q 8–12 h) and amoxicillin/clavulanic acid (13.75 mg/kg q 12 h). Instructions to feed only canned or softened kibble and to avoid toys or mouth play were given to the owners.At a 14-day post-operative recheck examination the patient was doing well with occasional sneezing. The extraoral closure had healed well. The intraoral incision was mostly healed with only a pin-point (~0.5 mm) dehiscence seen at the rostral extent. At the next recheck under general anesthesia another 14 days later, persistence of the pin-point oronasal fistula previously noted was identified. Surgical correction was declined by the owners, as the patient was asymptomatic. Two weeks later, the patient presented for left nasal discharge with suspected rhinitis suspected to be due to the persistent pin-point oronasal fistula. Medical management was initiated with azithromycin . At the last contact with owners, five months post-operative, the patient was doing well with no sneezing or nasal discharge and no gross evidence of recurrence.A 13-year-old neutered male Labrador retriever was referred to the Radiation Oncology service for evaluation and treatment of a suborbital spindle cell sarcoma. On presentation, a 1.5 cm diameter, multilobulated mass could be appreciated intraorally at the palatal aspect of the right maxillary second molar tooth. Thoracic radiography and regional mandibular lymph node aspiration was performed prior to referral, which revealed reactive lymphoid tissue and no evidence of pulmonary metastasis or intrathoracic lymphadenopathy. Head CT revealed a 3 cm x 2 cm soft tissue attenuating mass along the right caudal maxilla extending from the distal aspect of the right maxillary second molar tooth and into the suborbital adipose tissues with enlarged and attenuating zygomatic salivary gland .The patient was referred to the Dentistry and Oromaxillofacial Surgery service for excision of the OZMC and associated musculature, oral mucosa, gingiva and dentition utilizing a combined intra- and extraoral approach with a transpalpebral exenteration. The excision extended from the furcation of the right maxillary fourth premolar tooth and extended caudally to the mid-zygoma. From dorsal to ventral, the excision extended from the inferior orbital crest of the lacrimal bone and extended into the mandibular ramus and body just ventral to the mandibular second and third molar teeth. The excision including portions of the caudal maxilla, lacrimal, frontal, palatine, zygomatic, temporal, pterygoid and mandible bones as well as the zygomatic salivary gland. The palatal extent of the excision was the median palatine raphe. Following excision, the mesial and palatal roots of the maxillary fourth premolar tooth were extracted and the wound was closed as described above.Histological evaluation of the resected tissue confirmed the diagnosis of spindle cell sarcoma, but could not differentiate the tumor between fibrosarcoma and nerve sheath sarcoma. Greater than 1 cm tumor free histologic margins were reported. The dog recovered uneventfully from general anesthesia without any need for blood products and was managed overnight in the CCU on hydromorphone (0.5 mg/kg q 6 h IV). Meloxicam (0.2 mg/kg SQ) and ampicillin (22 mg/kg IV) were also administered. The dog was discharged 48 h post-operatively with oral acetaminophen (11 mg/kg q 8 h), gabapentin (10 mg/kg q 8 h PO) and meloxicam (0.1 mg/kg q 24 h). Instructions to feed only canned or softened kibble and to avoid toys or mouth play were given to the owners.Staphylococcus pseudointermedius (MRSP). The patient was instructed to stop the prescribed amoxicillin/clavulanic acid and was provided a 2-week course of oral doxycycline (6 mg/kg q12 h) and metronidazole (10 mg/kg q12 h) as per the results of sensitivity testing. The patient presented at 23 weeks post-operatively for definitive surgery to close the oronasal fistula. An island axial pattern flap of the left hard palate was utilized to close the previously described defect. The patient was discharged with oral meloxicam (0.1 mg/kg q24 h) and gabapentin (13 mg/kg q8 h) for 14 days. The patient presented 4 weeks after revision surgery reportedly doing well at home. Oral examination confirmed healing of the previously repaired oronasal fistula site. Exposed palatal bone that was left to heal by second intention showed minor signs of granulation. Concern for inhibition of healing from the previously diagnosed MRSP was communicated to the client and the patient was restarted on the previous antibiotic regimen. The patient was reassessed by his primary care practitioner 5 weeks after revision surgery and photographs confirmed a near complete granulation bed at the open site. Follow up email contact with the client 13 weeks after revision surgery confirmed the patient was doing well at home with complete healing of the surgery site.The patient returned 2 days post-operatively and a small amount of mucoid discharge from the rostral skin incision was noted. The client was instructed to give oral amoxicillin/clavulanic acid (13.75 mg/kg q 12 h). At the time of oral surgery recheck and skin suture removal 14 days post-operatively the owners reported the dog was doing very well at home, eating and drinking well and was not experiencing difficulty breathing. However, occasional “hacking” had been observed. The extraoral closure site and the rostral extent of the oral incision was healed. However, an ~2 cm diameter area of dehiscence was noted at the caudal most extent of the intraoral closure site. The owners were advised that a definitive repair of the resultant oronasal fistula would be delayed until inflammation in all tissues had resolved. At 13 weeks post-operatively the client reported the development of a purulent draining tract on the face at the previous surgical site. Physical examination of the patient at 14 weeks post-operatively confirmed a purulent draining tract at the level of the medial canthus of the exenteration site. At 16 weeks post-operatively the patient was anesthetized for examination, which revealed communication of the facial draining tract with the previous surgery site and a concurrent oronasal fistula. Samples were taken from the draining tract for culture and sensitivity prior to thorough lavage and debridement. The patient was discharged with instructions to continue the previously prescribed amoxicillin/clavulanic acid and meloxicam pending culture and sensitivity results, which yielded moderate growth of methicillin resistant An 8-year-old spayed female vizsla was referred to the Medical Oncology service for evaluation of an osteosarcoma located inferior to the right eye. The referring veterinarian had previously extracted the right maxillary fourth premolar tooth and the right maxillary first molar tooth at the time of biopsy. On presentation, the visible mass measured 2.7 cm in length and was firm and non-mobile. Fine needle aspirates of the mandibular lymph nodes revealed no evidence of local metastasis. Thoracic CT revealed a partially mineralized 0.4 cm nodule in the left cranial lung lobe of unknown significance. Head CT revealed a 3.2 cm × 3.2 cm × 3.5 cm osteolytic and osteoproliferative soft tissue and mineral attenuating, moderately contrast enhancing mass within the right caudal maxilla. The mass extended caudally into the right pterygopalatine recess and orbital space causing dorsolateral displacement of the right globe and caudoventral displacement of the right zygomatic salivary gland. The mass extended rostrally and ventrally to include the alveolar bone associated with the previously extracted right maxillary fourth premolar tooth.The case was referred to the Dentistry and Oromaxillofacial Surgery service for surgical treatment. The tumor was resected as an en bloc excision of the OZMC and associated musculature, oral mucosa, gingiva and dentition utilizing a combined intra- and extraoral approach with a transpalpebral exenteration. The osseous excision extended from the furcation of the right maxillary second premolar tooth to the mid-zygoma. The medial extent of the excision was the orbital part of the frontal bone and 2 mm to the contralateral side of the median palatine raphe. The excision including portions of the caudal maxilla, lacrimal, frontal, palatine, zygomatic, temporal, and pterygoid bones. Following excision, the wound was closed as described above.The dog recovered uneventfully from general anesthesia and was managed overnight on CRI of fentanyl (2–5 μg/kg/hr) and ketamine (2–4 μg/kg/min) and an injection of carprofen (4.4 mg/kg SQ). A transdermal fentanyl patch (75 μg/hr) and piperacillin/tazobactam (45 mg/kg IV q 8 h) was also administered. The dog was discharged the following day with oral gabapentin (13 mg/kg q 8 h) and carprofen (4.4 mg/kg q 24 h). A nylon stockinette, and a loosely fitted tape muzzle was also applied to limit oral range of motion and tension on the intraoral surgery site. Instructions to feed only canned food and to avoid mouth play were given to the owners.Histological examination confirmed the diagnosis of osteosarcoma with a narrow margin (500 μm) at the caudal aspect of the excision. Four days post-operatively, the patient presented for evaluation of malodorous smell from the oral cavity and hyporexia. Examination revealed a 6 cm dehiscence at the most caudal aspect of the intraoral closure. There was also purulent discharge present at the extraoral closure. The patient was discharged with instructions to continue medications as previously prescribed. The client reported unwillingness to administer gabapentin due to the level of sedation of the patient. Amoxicillin/clavulanic acid (17 mg/kg PO q 12 h) and metronidazole (10 mg/kg PO q 12 h) was prescribed, empirically. Mirtazapine (0.7 mg/kg PO q 24 h) was also prescribed as an appetite stimulant. Oronasal fistula repair was planned to occur when local inflammation and infection had resolved.The patient was assessed by a veterinary dental specialist at a separate institution 7 days post operatively where physical examination revealed dehiscence of the extraoral closure and complete dehiscence of the intraoral closure resulting in a maxillo-oronasal fistula. Given the nature of the dehiscence and the likelihood of pursuing radiation therapy, surgical repair was recommended ahead of the previously planned schedule.Fourteen days post-operatively the patient presented for repair of the maxillo-oronasal fistula with the Dentistry and Oromaxillofacial Surgery service. Under general anesthesia, a right inferior alveolar nerve block was administered with 1.5 mg bupivacaine and 15 μg buprenorphine. The right mandibular molar teeth were routinely extracted to prevent occlusal trauma to the maxillary soft tissue. Samples from the fistula were collected and submitted for culture and sensitivity. The maxillo-oronasal fistula was thoroughly lavaged. A myofascial axial pattern flap based on the temporalis muscle was utilized for repair of the oronasal fistula as described by Cavanaugh et al.  The patiSignificant facial edema developed post-operatively, which resolved by the fourth post-operative day. However, the patient was intermittently anorexic and the owners were unable to provide medications orally. The patient was re-admitted to the hospital 8 days following the revision surgery. An area of eschar overlying the rostral aspect of the extra-oral incision was debrided under sedation revealing a 3 cm diameter cutaneous dehiscence with exposure of the underlying temporalis muscle. The wound was thoroughly lavaged and cleansed with dilute chlorhexidine and sterile saline. Nylon  sutures were placed around the periphery of the wound to allow for a tie-over bandage to be placed. An absorbable, antimicrobial polymeric wound matrix was used for direct wound dressing followed by sterile gauze and a moisture-protective barrier superficially. The bandage was held in place with umbilical tape. An esophagostomy tube was routinely placed during a short general anesthetic event the day prior to discharge to allow for administration of all oral medications. The patient was discharged with a new transdermal fentanyl patch (75 μg /hr) and oral carprofen (2.2 mg/kg q 12 h), acetaminophen (10 mg/kg q 12 h), amoxicillin/clavulanic acid (17 mg/kg q 12 h), marbofloxacin (5 mg/kg q 24 h) and ondansetron (2 mg/kg q 12 h). All medications were instructed to be administered via the esophagostomy tube, but the patient was to be allowed and encouraged to eat canned food. The owner was instructed to change the tie-over bandaging materials every 3 days.The patient re-presented to the Dentistry and Oromaxillofacial Surgery service 20 days after the revision surgery. Orofacial examination revealed a layer of healthy granulation tissue over the dermal dehiscence site. Additionally, there was approximately 80% epithelialization of the intra-oral myofascial flap and no evidence of a persistent oral nasal fistula on conscious oral exam. The esophagostomy tube was removed and the sutures previously placed for the tie-over bandage were also removed. Surgical closure of the extra-oral dehiscence was discussed with the owner, but healing by second intention was elected. A soft food diet was recommended for an additional 3 weeks.Six weeks following revision surgery, the patient presented to the medical oncology service to discuss adjunctive therapy options. Moderate restricted range of motion was identified when opening the patient's mouth. The patient was placed under general anesthesia for thorough oral examination and head CT which revealed a communication between the oropharynx and nasopharynx measuring ~1 cm in diameter, which was located at the junction of the myofascial flap and the soft palate. The client reported no clinical signs associated with the fistulation. Given the non-clinical nature of the fistula, surgical repair was not pursued. With narrow histologic tumor margins and the pulmonary nodule identified on thoracic CT, radiation therapy and chemotherapy were recommended, but not pursued by the owner.In the report conveyed here, we have described a surgical technique to resect extensive OZMC tumors in which an intraoral approach is combined with a horizontal extraoral incision along the dorsolateral muzzle and zygoma that also incorporates a transpalpebral approach to orbital exenteration. This approach is a modification of a previously described combined intra- and extra-oral approach in order to access tumors of the caudal maxilla, in which the extraoral incision is made ventral to the globe .Excision of the extensive OZMC tumors in this series included the entire inferior orbit and significant portions of the medial orbit. The medial orbit osteotomy was generally along a line beginning rostrally in the orbital crest of the lacrimal bone, just superior to the fossa of the lacrimal sac; coursing caudally along a line just inferior to the inferior orbital crest and optic canal; terminating just caudal to the hamulus of the pterygoid bone. The angle of approach (~40° from the median plane) for the medial orbitectomy must also be exact to ensure communication with the intraoral palatal approach and the caudal nasopharynx rather than penetrating into the rostral cranial fossa. An alteration of as little as 15° would direct the saw blade into the cerebrum, which could lead to significant neurological complications –14 Figu.In addition, maintaining the globe without orbital reconstruction may result in enophthalmos and resultant significant ocular complications . DiplopiVisualization is vital to successful complete excision of OZMC tumors. Previous literature indicates that invasive orbital and periorbital tumors are often inadequately excised with local recurrence or persistence occurring in 36.7% of cases . In ordeExenteration as part of the surgical plan for these patients was further supported by the possible need for post-operative radiation therapy. Commonly reported ocular-related radiation side effects are conjunctivitis, alopecia, keratoconjunctivitis sicca, erythema, uveitis and ulceration –18. Keraonly, not to treat microscopic disease after surgery. Stereotactic radiation therapy is not recommended following surgery due to the concern of high doses of radiation to normal tissues especially to nerves, bone, and ocular structures. In the population of patients discussed here, definitive radiation therapy would be recommended following surgery.The decision to exenterate the globe may be debatable. Highly conformal intensity modulated and image guided forms of radiotherapy have been successfully utilized to spare the globe from severe radiation toxicity in the setting of full-course definitive-intent radiotherapy in dogs with maxillofacial tumors. On the other hand, at our institution, stereotactic radiation therapy is selected on a case by case basis and utilized in the gross disease setting In the future, we may consider sparing the globe in the few cases in which orbital reconstruction is pursued and exenteration is not necessary to achieve a medial orbitectomy. Orbital reconstruction would be necessary to avoid complications related to diplopia. Orbital reconstruction showed promising results in one case series . Short-tThe major complication seen in these patients was significant intraoral incision line tension, which led to dehiscence and persistent oronasal fistula in three out of the five cases. The tension seen in these patients was not unexpected given that a significant amount of alveolar and palatal mucosa was excised as part of the excision. Tension was also related to the medial extent of removed tissue, often to or across the median palatine raphe. Taken with limited buccal mucosa, the lack of palatal mucoperiosteum made the reconstruction of an oral vestibule quite challenging. The dehiscence rate in this small case series (60%) is higher than previous reports in maxillectomies of 5–33% , likely In order to improve visualization and access to the caudal oral cavity, a commissurotomy was also performed. It is conceivable that the proximity of the commissurotomy to the intra-oral mucosal incisions may have compromised the mucosal vascular supply and contributed to the risk of dehiscence. It was determined that the ability to achieve clean margins through optimal visualization was of paramount importance. As no patient experienced visible evidence of post-operative flap necrosis, the author's do not believe vascular supply was compromised. However, should one wish to use an angularis oris mucosal axial pattern flap to aid in intraoral closure, a commissurotomy would not be possible as it would likely damage the angularis oris artery. A plausible alternative would be to make a full-thickness, vertically oriented releasing incision through the superior labia to connect with the most rostral extent of the extra-oral incision; in effect, creating a full thickness labial flap that could be reflected caudally. This approach has been used for visualization of the OZMC in human oromaxillofacial surgery  and has While all our patients generally had minimal intra-operative hemorrhage, two patients did require a transfusion. Previous reports describe significant hemorrhage as the most common intra-operative complication seen in maxillofacial surgery . HemorrhThe surgical approach described here in which an orbital exenteration is used in conjunction with the dermal incision of a combined intra-and extraoral approach provides significant benefits for the excision of extensive OZMC tumors. Excellent visualization is obtained, common intra-operative complications are minimized and post-operative ocular complications are eliminated. The major complication, primarily caused by the size of the excision rather than the approach, is intra-oral wound dehiscence. This complication may be mitigated with the addition of axial pattern flaps  to avoid intra-oral wound tension.The original contributions generated for the study are included in the article/supplementary material, further inquiries can be directed to the corresponding author/s.Ethical review and approval was not required for the animal study because the surgical procedures reported within this manuscript were performed in the course of providing a medically necessary procedure. The results are retrospective in nature and included clinical cases. Therefore, ethical review and approval was not required and is exempt from IACUC requirements. Written informed consent was obtained from the owners for all procedures performed.AT, BR, and AG: drafting of case descriptions and discussion and final approval of version to be published. AT and JS: overall drafting of entire manuscript. JS: surgical concept and design, graphic development, interpretation, manuscript drafting, and final approval of version to be published. 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.


Malignant mesothelioma of the tunica vaginalis is a rare tumour which comprises less than 1% of all mesotheliomas.69-years old patient with painful hard mass and hydrocele in the right scrotum to whom a right hydrocelectomy was performed. Any history of scrotal trauma or exposure to asbestos was not present. Excisional biopsy revealed a multinodular tumour with focal areas of necrosis and infiltrative growth. According to morphological and immunohistochemical findings, diagnosis of malignant biphasic mesothelioma of the tunica vaginalis testis was made. Two months after hydrocelectomy, right inguinal orchidectomy was performed. Post-surgical whole body CT scan revealed paraaortic and pararenal lymphadenopathy, likely to be metastatic. Adjuvant treatment with 6 cycles of cisplatin and pemetrexed was applied. After 3 cycles of chemotherapy, CT scan showed progression and the treatment was changed to gemcitabine 1 month after.Although malignant mesothelioma of the tunica vaginalis is a rare malignancy, it poses a diagnostic challenge which can mimic common inguinal or scrotal diseases such as hydrocele. Despite aggressive surgical procedures or adjuvant therapies, the prognosis remains poor. Malignant mesothelioma of the tunica vaginalis testis  is a rare tumour, comprising less than 1% of all mesotheliomas . Since tA 69-year-old man was referred to our hospital after the right hydrocelectomy when suspicious, non-homogeneous tissues were found intraoperatively. Ultrasound imaging and computed tomography (CT) scan were not performed before the surgery. On physical examination, right scrotum appeared to be edematous, hard and painful mass was evident with no palpable lymphadenopathy. Mentioned symptoms have been observed for a year. The patient denied any history of scrotal trauma or exposure to asbestos. 8 years ago he was diagnosed with prostate adenocarcinoma (Gleason 3 + 3 = 6) cT1cN0M0 stage II, the result of external beam radiotherapy (74 Grays / 37 fractions) treatment was radical. One year due to PSA (prostate specific antigen) relapse, there were androgen deprivation therapy prescribed. 9 months thereafter androgen deprivation therapy was discontinued due to low PSA rates. To the date of testicular mesothelioma diagnoses serum PSA level was 0.1 ng/ml. No other comorbidities were reported to this patient. Levels alpha-fetoprotein and beta human choriogonadotropin were not elevated. The excisional biopsy revealed a multinodular tumour with focal areas of necrosis and infiltrative growth. Mixed epithelial and spindle cell proliferation was present. The bulk of the epithelial component contained confluent cords and nests of monotonous, ovoid to polygonal cells with pale or eosinophilic cytoplasm (Fig. Two months after hydrocelectomy, right inguinal orchidectomy was performed. Paratesticular space was largely replaced with yellow-white nodular masses with cystic spaces Fig. . The subMalignant mesotheliomas are uncommon tumours, which can develop from the coelomic epithelium at the pleura, peritoneum, pericardium and tunica vaginalis testis. Mesothelioma of the tunica vaginalis testis represent only 0.3–5% of all mesothelial neoplasms . AccordiTesticular mesotheliomas usually occur between 55 and 75 years . The incExposure to asbestos is a major risk factor for the development of pleural and peritoneal mesotheliomas. However, asbestos link to tunica vaginalis tumours is not well elucidated , 7, 8. IOther suspected risk factors for the disease include trauma, herniorrhaphy, long term hydrocele or spermatocele, long-term epididymitis, orchitis or other inguinal inflammation , 10–14. Therapeutic radiation for other malignancies is a well-established risk factor for pleural and peritoneal mesotheliomas , 17. TheThe rarity of testicular mesothelioma poses challenges to its diagnosis and that is rarely achieved preoperatively. Main symptoms are nonspecific and patients usually present with enlargement of the scrotum, recurrent hydrocele and palpable scrotal mass . CommonlIn typical cases tunica vaginalis mesotheliomas resemble their pleural and peritoneal counterparts . Pure epThere is scarce evidence regarding treatment of this disease. Therapeutic principles are frequently extrapolated from management of pleural or peritoneal mesotheliomas and may feature a combination of surgery, radiation therapy and chemotherapy . In our In summary, we present a case of a patient with testicular mesothelioma of tunica vaginalis. The rarity of testicular mesothelioma poses challenges to its etiology research, diagnosis and treatment. Diagnosis of testicular mesotheliomas is challenging, as the tumour lacks specific clinical and radiologic features, and the reported sex cord-like pattern proves its histological diversity. Despite aggressive surgical procedures or extratesticular mesothelioma-based adjuvant therapies, the prognosis remains poor.


A radical prostatectomy was performed, and the ensuing follow-up was unremarkable with no signs of recurrence. Fourteen years later, the patient presented with an obstructive pyelonephritis due to a left-sided ureteral stone requiring ureteral stenting. An exophytic tumor was seen in the lining of vesicourethral anastomosis and surgically excised after the pyelonephritis subsided. The histopathological and immunohistochemical analysis revealed a ductal cancer of the prostate consistent with a late local recurrence. Serum PSA was below the limit of detection. Re-staging performed by an MRI of the pelvis, thoracoabdominal CT scan, and gallium-68 PSMA-PET did not reveal any other signs of disease. The ensuing follow-up is planned with regular flexible cystoscopy and computed thoracoabdominopelvic CT scans.We report the case of a 74-year-old patient in whom a ductal prostate cancer was incidentally endoscopically diagnosed in the course of ureteral stenting due to a left distal ureteral stone. The initial PSA was 0.8  The majority of prostate cancers are adenocarcinomas, with histological variants seen in 5–10% of carcinomas . CompareIn localized disease, radical prostatectomy or external beam radiation therapy is recommended. Optimal management poses a challenge for clinicians, as ductal adenocarcinomas may not express PSA, potentially delaying the diagnosis of disease and complicating follow-up.The at-the-time 60-year-old patient was referred to our clinic 14 years ago due to persistent left-sided flank pain for over six months. The patient described intermittent radiating pain to the left groin and denied micturition disorders such as urinary discomfort or gross hematuria. Left-sided hydronephrosis was seen on renal ultrasound, and a kidney, ureter, bladder (KUB) plain film radiography was performed demonstrating an 8 mm large opaque structure in projection of the distal ureter, suspicious for a ureteral stone as well as a 10 mm large bladder stone. Stone therapy with transurethral lithotripsy and ureteral stenting on the left was planned. During cystoscopy, an exulcerating lesion with whitish, fibrinous necrotic slough was seen left of verumontanum, with no other lesions apparent. The bladder stone was disintegrated and removed using a grasping forceps, and a biopsy of the tumor was performed. The ureteral stone was pushed back into the left renal pelvis and a double-pigtail stent was inserted. Further stone treatment was performed by extracorporeal shock wave lithotripsy with removal of the ureteral stent after radiological disintegration of the stone was seen.μg/l. The tumor markers carcinoembryonic antigen (CEA), carbohydrate antigen 19-9 (CA 19-9), and cancer antigen 125 (CA 125) were normal. Staging was conducted with a pelvic MRI, thoracoabdominal CT scan, and bone scintigraphy without evidence of metastasis. A radical prostatectomy with extended pelvic lymphadenectomy was performed, with ductal adenocarcinoma seen in both prostate lobes without extraprostatic extension, nodal metastasis, or positive surgical margins, corresponding to a pT2c, pN0, cM0, Gleason score 7b (4 + 3), R0 stage. The postoperative course was uneventful, and the patient was subjected to regular clinical follow-up with digital-rectal examination and serum PSA control. Serum PSA was not detectable in the first postoperative control and remained below the limit of detection throughout the whole duration of follow-up. The patient had persistent postoperative stress urinary incontinence, which required the use of 1-2 pads per day with an otherwise unremarkable follow-up.The histopathological analysis revealed a ductal adenocarcinoma of the prostate. Digital rectal examination of the prostate was not suspicious, and total serum PSA was 0.8 Fourteen years after the initial diagnosis, the now 74-year-old patient was admitted to the emergency department with fever and left-sided flank pain. A computed tomography of the abdomen revealed an 8 mm stone in the distal left ureter with consecutive dilation of the left ureter and perirenal stranding of the left kidney indicating obstructive pyelonephritis. Intravenous antibiotic therapy was initiated, and the immediate ureteral stenting of the left ureter was performed with a double-pigtail stent. During cystoscopy, a solitary 15 mm large tumorous lesion was seen on the left side of vesicourethral anastomosis, which was not resected due to the urosepsis. The patient quickly recovered from the infection and was subjected to a transurethral resection of the tumor and semirigid ureteroscopy for stone removal. The tumorous lesion of the bladder was resected in toto and the ureterolithiasis successfully removed.The histological analysis revealed a ductal adenocarcinoma consistent with a local recurrence of the known ductal adenocarcinoma of the prostate . ImmunohImaging with a pelvic MRI, thoracoabdominal CT scan, and a gallium-68 PSMA-PET scan revealed no signs of metastasis. The case was discussed at our multidisciplinary tumor board, and external beam radiation therapy of the prostate bed was recommended to reduce the risk of recurrence. The patient wished to abstain from further therapy so as not to worsen the preexisting stress urinary incontinence. A flexible cystoscopy was performed 3 months postoperatively demonstrating an unremarkable vesicourethral anastomosis. Further follow-up is scheduled with annual cystoscopies and thoracoabdominopelvic CT scans.In the reported case, a very late local recurrence was incidentally detected fourteen years after initial diagnosis. Prior to that, regular follow-up had been performed with digital rectal examinations and serum PSA detection, with no signs of local recurrence and a serum PSA which was below detection level. Furthermore, the Ki67 proliferation index of the resected local recurrence was very low at 3%, indicating a very slowly growing disease, which may partially explain the long latency period between primary diagnosis and recurrence.As with the initial diagnosis, the recurrence was only detected due to left-sided ureterolithiasis. Chung et al. performed a retrospective population-based case-control study to assess the association of prostate cancer and urinary calculi. While they were able to show that the incidence of prior urinary calculi was higher in patients with newly diagnosed prostate cancer compared with the control group 21% vs. 14.1%), no significant relationship was seen in regard to ureter calculi [.1%, no sThe treatment of localized disease of ductal adenocarcinomas of the prostate consists of radical prostatectomy or external beam radiation therapy. When compared with acinar adenocarcinomas of the prostate, ductal cancers were more likely to present with advanced disease and a lower PSA indicating the challenging diagnosis . PatientDuctal adenocarcinomas of the prostate are rare histological variants and should be treated as a separate tumor entity with an individualized management. When localized disease is present, a radical prostatectomy or external beam radiation therapy can be performed. The further follow-up has to be tailored to the individual patient, as PSA is not a reliable tool for assessing the existence of disease. Furthermore, regular thoracoabdominopelvic imaging, for instance, with regular CT or MRI scans as well as flexible cystoscopies should be considered.


Ectopic pheochromocytomas, the incidence of which is >15%, can occur throughout the entire body but seldom on the diaphragm. Surgery may the first-choice treatment for ectopic pheochromocytomas.We herein describe a 61-year-old woman with an atopic diaphragmatic pheochromocytoma. She had a 7-year history of paroxysmal headaches, palpitations, and hypertension with no obvious causes; these symptoms were alleviated by nifedipine and metoprolol. Computed tomography (CT) revealed a slightly hypodense lesion on top of the right hepatic lobe. A Metaiodobenzylguanidine (MIBG) scan showed increased radioactive in the lesion. After adequate preoperative preparation, we removed the mass. During the operation, we found that the mass was located on the diaphragm. The pathological examination showed that the main pathologic change was paraganglioma. The patient recovered well after surgery with no recurrence of her hypertension, palpitation, or headache.Diaphragmatic pheochromocytoma is a rare kind of ectopic pheochromocytomas, which can also affect the patient’s quality of life. Combination of qualitative and positioning tests can assist in diagnosis of ectopic pheochromocytoma. Surgical resection is an effective treatment method. These tumors can occur throughout the entire body, especially the para-aortic region, renal hilus, and inferior vena cava. They are relatively rare on the base of skull, heart, gastrointestinal system, sacroiliac blood vessels, ureter, uterus, and ovaries . To our 2A 61-year-old woman presented to our institute for evaluation of a liver mass. She had a 7-year-history of paroxysmal headache and palpitation. During a routine physical examination, an approximately 3-cm liver mass had been found by ultrasonography, and a hepatic hemangioma was suspected. She reported intermittent symptoms of headache, palpitation, and hypertension up to 200/110 mmHg with no obvious causes since 2007. These symptoms were alleviated after taking nifedipine and metoprolol. The symptoms thereafter resolved, and she stopped the treatment herself. In September 2013, the symptoms recurred and were frequently aggravated with postural hypotension. The patient had a 7-year history of diabetes mellitus, a 3-year history of cholecystolithiasis, and a 6-month history of hyperlipidemia. She presented to our hospital for further diagnosis and treatment. Physical examination revealed a body temperature of 36.5 °C, pulse of 70 bpm, standing blood pressure of 120/80 mmHg, and supine blood pressure of 140/100 mmHg. Cardiopulmonary and abdominal examinations showed no obvious abnormalities.Considering that these symptoms are consistent with pheochromocytoma, we ran many tests. The patient’s blood norepinephrine level was high, and her 24-h urine epinephrine, norepinephrine, and dopamine levels were 2.66, 63.94, and 130.54 μg, respectively. Thoracic and abdominal enhanced CT  revealedOur presumptive diagnosis was liver paraganglioma based on the clinical manifestations and test results. We initiated treatment with phentolamine at 5 mg twice a day; this was gradually increased to 27.5 mg/day. We monitored the patient’s erect and supine blood pressure, heart rate, weight, and intake–output fluid volume. After initiating treatment, her blood pressure stabilized and she gained 6 kg of body weight. We then performed an operation on 10 June 2014. During surgery, we found that the tumor was protruding from the diaphragm. The tumor had a diameter of 4 cm, and soft pitting was present on top of the liver. A small amount of yellow moss was stuck to the external surface of the soft/solid tumor, which was vascularized. The patient’s blood pressure increased when the tumor was touched. We sliced the diaphragm with a 0.5-cm margin around the tumor by electrocoagulation and sewed the wound closed. Before suturing the diaphragm, we filled her lung to exhaust the air in the chest and checked in water. We placed a drainage tube under the diaphragmatic wound and returned the patient to the intensive care unit. The patient recovered well; her symptoms of paroxysmal hypertension and palpitations disappeared, and the catecholamine levels in her blood and urine returned to normal.The final pathological examination  suggesteSeveral weeks before the present writing, she underwent outpatient follow-up and was still clinically well without recurrence.3The incidence of ectopic pheochromocytoma is >15%, and both the location and clinical presentation are variable . ConsequThe typical clinical manifestations of pheochromocytoma are paroxysmal hypertension, headache, palpitations, and excessive perspiration . In the The most difficult aspect of the diagnosis of pheochromocytoma or paraganglioma is the positioning diagnosis. CT, magnetic resonance imaging, and MIBG scans have great value in the locative diagnosis ,10. In pSurgery may be the only way to cure this disease . PreoperThe main risk in the postoperative period is hypotension ,10. Thus4In conclusion, Diaphragmatic pheochromocytoma is a rare kind of ectopic pheochromocytomas, which can cause various symptoms and affect the patient’s quality of life. A qualitative diagnosis can be made by the presence of typical symptoms combined with blood and urine examination of catecholamines. The positioning diagnosis can be performed by medical imaging examinations such as CT and especially MIBG scan. Surgical resection is an effective treatment method, but it requires adequate preparation before surgery, strict intraoperative monitoring, and careful management after surgery.None.None.The study is exempt from ethnical approval in our institution.Written informed consent was obtained from the patient for publication of this case report.Xiangan Wu: Data curation, Writing - Original draft.Bao Jin: Investigation.Shi Chen: Resources.Shunda Du: Writing - Review & Editing, Project administration.Yilei Mao, Xinting Sang: Supervision.Not acquired.The correspondent author.Not commissioned, externally peer-reviewed.


Myopericytomas are exceedingly rare soft-tissue tumors with less than 10 cases including radiological depictions. We report three new cases of benign myopericytomas located in the soft-tissues of the hand in adult patients. A pre-treatment MRI was available for all patients and systematically evidenced well-defined, lobulated tumors closely related to the superficial palmar vascular arch and/or digital vessels with a perivascular pushing growth pattern that correlated with pathological findings. Though rare, this small case series show that myopericytomas display recurrent imaging features that could support their radiological diagnosis. A 72-year-old male without significant medical history presented with a lump of the right palmar hand side. This lesion was slowly growing for more than 5 years and became progressively painful.T2 weighted imaging (T2WI) and low SI on T1WI, similar to the muscles. There was no cystic, fatty, necrotic, myxoid, or fibrotic component. The surrounding tissues did not exhibit edema, aponeurotic or peritumoral enhancement. The tumor showed a limited intratumoral heterogeneity on T2WI due to intratumoral nodules delineated by lower SI on T2WI.On MRI, the tumor measured 54 mm and was well-circumscribed with lobulated margins. It was closely related to the superficial palmar vascular arch with perivascular nodular clusters demonstrating a pushing growth pattern. The tumor showed thinner extensions following the common palmar digital vessels of the fifth finger . It dispThe close relationship to palmar vessels raised the hypothesis of a tumor of vascular or pericytic origin such as hemangiopericytoma, angioleiomyoma, myofibroma or myopericytoma. The slow progression, the well-defined margins, the lack of necrosis and the limited heterogeneity were rather in favor of a benign lesion but soft-tissue sarcoma, especially leiomyosarcoma, could not be excluded.An ultrasonography-guided core needle microbiopsy was performed because the tumor was deep-seated, over 5 cm without obvious diagnosis. The histopathological analysis revealed a proliferation of cells containing significant eosinophilic cytoplasm and arranged in short fascicles circumscribing tightly small blood vessels. There was no cytologic atypia and the proliferative activity was low (1–2% of cells positive for Ki67). Immunohistochemistry demonstrated a strong and diffuse positive staining for alpha-smooth muscle actin, desmin and h-caldesmon. These features were compatible with a benign myopericytoma of the hand.The patient was subsequently treated with a curative surgery. The gross examination of the surgical specimen showed strong analogy with MRI. The tumor was well-circumscribed, multinodular and solid with a yellow-white pearl color. The ‘hemangiopericytic’ concentric perivascular growth pattern was responsible for a thinning of the vessel lumen . No infaFollowing these findings, we performed a request on the pathological databases of two French Sarcoma Reference Centres and we identified a total of 13 patients with myopericytomas of the upper limb including two with a pre-treatment MRI. Interestingly, they also shared this perivascular pushing growth pattern.The first patient was a 30-year-old female. She presented with a discrete lump of the palmar and ulnar side of the left wrist that became progressively painful with paraesthesia in the ulnar nerve territory. A MRI was performed showing a 45-mm-long, well-circumscribed, lobulated and multinodular tumor that followed the same orientation as the ulnar vessels and spread along the superficial palmar vascular arch with small, thin extensions following the common palmar digital vessels towards the fifth, fourth and third fingers . After iT1WI, high SI on fat sat T2WI and homogeneous contrast-enhancement  was only available for Patient 2 in order to guide the biopsy—the other ones being performed out of our sarcoma reference center and not available on our PACS.Regarding the distal upper extremities, less than 25 cases have been published so far, including less than 10 with imaging. However, they share common features that could help to establish diagnosis.T1WI, high SI on T2WI, with a discrete heterogeneity due to an intratumoral nodular aspect, and rather lobulated contour. The sizes ranged from 9 to 54 mm. The previous studies systematically stressed the proximity to an artery or a vein. Interestingly, myopericytomas seemed to encase and push adjacent organs rather than invade them. After contrast agent injection, myopericytomas constantly demonstrated a strong, avid enhancement related to their vascular origin, without peritumoral enhancement. Interestingly, Van Camp et al performed an arteriography and a dynamic-contrast enhanced MRI of a myopericytoma showing an arterial enhancement and a washout.7 Such a kinetic of enhancement may be reminiscent of a glomus tumor and could help making diagnosis.MRI is the best imaging modality for characterizing myopericytomas. In all reports, myopericytomas displayed iso- to low SI on 1,2,8 Immunohistochemistry systematically found strong and diffuse positive staining for ASMA and h-caldesmon, while staining for CD34 and desmin seemed more inconstant.8 Interestingly, myopericytomas are underlined by somatic mutations of PDGFRB (Platelet derived growth factor receptor B—a growth factor involved in angiogenesis) in about 60% of cases. Nevertheless, it cannot help to distinguish them from other pericytic tumors because PDGFRB alterations can also be found in angioleiomyomas and myofibromas.9,10In the three present cases, the final diagnosis was rendered by histopathological analysis. Hematoxylin and eosin stained slides demonstrated the typical features associated with myopericytomas and notably a concentric perivascular growth pattern. This appearance is in keeping with the pericytic origin of these tumors encasing the vascular structure from which they arise and sometimes protruding into the lumens of the vascular branches .1,2,8 Im11 Six of these patients developed metastasis in the liver, skin, heart, brain and bone. Three of them died of their disease.To the best of our knowledge, 11 cases of malignant myopericytomas have been reported. They were histopathologically characterized by high mitotic rate, hypercellularity, pleomorphism and necrosis but they lacked radiopathological correlations.The treatment for benign myopericytomas consists of a curative surgery with rare local recurrence likely due to incomplete resection, thereby requiring regular follow-up based on clinical examination.To conclude, myopericytomas belong to a continuous spectrum of tumors of pericytic lineage with nodular, concentric, perivascular pushing growth pattern that parallels their imaging features. Myopericytomas of the hand typically present as well-circumscribed and nodular lesions attached to (or encasing) the superficial palmar arch and its digital branches, together with an avid contrast-enhancement on MRI. Patients should be referred to a sarcoma reference center before surgery in order to perform imaging-guided core-needle biopsy because of potential malignancy.Myopericytomas are extremely rare ubiquitous tumours that have been recently described and are mostly encountered in the distal extremities of adults.They share common MRI features that correlate with histopathological analysis. They present as well-limited lobulated and nodular tumors with a close relationship to palmar vessels reminiscent of their pericytic lineage. They rather encase and push aside the vascular and other surrounding structures, instead of invading them.Patients with a suspicion of myopericytoma must be referred to a Sarcoma Reference Center because of potential malignancy.


Teaching Point: Aggressive angiomyxoma is a rare and benign soft tissue lesion, characterized by a “whorled” or “layering” appearance on T2-weighted and post-gadolinium magnetic resonance. A 49-year-old healthy woman presented to the hospital with a painless right gluteal swelling that progressively increased in size over the past two years and caused discomfort when cycling. No history of trauma was indicated.Rectal and vaginal examinations were normal. Laboratory findings, including tumor markers, were unremarkable.On ultrasound, a vascularized hypoechogenic and heterogeneous mass was found posterior to the ischio-pubic bone Figure .Magnetic resonance imaging (MRI) showed a mass in the right ischiorectal fossa, with well-defined limits and digitiform projections, measuring at least 6.8 × 5.8 × 3 cm. It shaped without invading adjacent structures, such as the right levator ani and the adductor muscles, the vagina, and the anal canal. The lesion showed iso-signal intensity to muscle on T1-weighted images Figure  and heteComputed tomography (CT) was consistent with MRI features, demonstrating an expansive lesion of low density with heterogeneous enhancement extending from the vulvar region to the right ischiopubic bone Figure .A core needle biopsy and subsequent surgical resection were performed, diagnosing an aggressive angiomyxoma (AA).The patient remains asymptomatic without imaging recurrence several months later.Aggressive angiomyxoma is a rare mesenchymal tumor with a predilection for the perianal and pelvic region. Characteristically, it’s a slow-growing benign tumor, with locally aggressive and infiltrative behavior. The majority of cases occur in premenopausal women, with a female-to-male ratio of 6:1. Patients frequently present with a painless and indolent growing mass, achieving large dimensions on presentation.MRI is the best imaging technique for characterization and extent assessment, having an essential role in surgical planning. Typically, the lesion demonstrates iso to low-signal on T1-weighted images and heterogeneous high-signal on T2-weighted sequences [On CT, AA is hypo or isodense and has variable enhancement after contrast .The differential diagnosis includes soft-tissue tumors with a myxoid pattern, such as angiomyofibroblastoma, soft-tissue sarcomas, pelvic myxoma, and a solitary fibrous tumor. These lesions don’t exhibit a whorled/layering appearance on MRI, a feature that contributes to differentiating them from AA .Complete surgical resection is the treatment of choice. Unfortunately, the infiltrative nature of AA is associated with difficult complete excisions, so that about 70% of cases recur in three years.


ENT specialists should never underestimate the clinical benign course of cervical masses both in adults and in young patients. ENT specialists should never underestimate the clinical benign course of cervical masses both in adults and in young patients. Lymph nodes affected by Castleman disease can mimic both benign and malignant disorders of the neck. Surgery can provide histopathologic diagnosis and cure with good prognosis for unicentric Castleman disease.A 20 years‐old woman referred to our evaluation for an asymptomatic slow‐growing cervical mass. The patient complained about the discomfort related to the esthetic appearance of the mass, and her past medical history was unremarkable. Magnetic resonance imaging (MRI) revealed an ovoid mass with peripheral contrast enhancement Figure . The patThe authors declare that there is no conflict of interest regarding the publication of this article.All authors contributed to the care of this patient. The authors wrote and critically reviewed the manuscript.


Castleman's disease (CD) is a primary lymphoproliferative disorder of the lymph nodes with rare extra-nodal primary affection. Presentation of case: Here we present a case of primary hepatic CD associated with hepatocellular carcinoma (HCC). Discussion: Sixty-two years old, male received direct-acting antiviral agents (DAAs) for HCV infection. Follow up revealed sustained virologic response; however, three hepatic focal lesions were accidently discovered. Triphasic CT confirmed the HCC nature of two masses while the other mass remained undiagnosed. Surgical intervention was the treatment of choice, and pathological examination showed a fairly circumscribed mass formed of angiolymphoid hyperplasia displayed atrophic germinal center, expanded mantle zone, and variable hyalinization. The radiological evaluation of lymph nodes was unremarkable. The patient is 40 months alive after resection, with no further management advised. Conclusion: The immune-modulatory effect of DAAs may induce hepatic CD development in a cirrhotic patient, necessitating further studies. A new radiologic finding was observed in the present case in the form of vessels traversing through the lesion with no attenuation or occlusion. Pathology remains the gold standard in the diagnosis of CD. •Eighteen cases of primary hepatic Castleman's disease (CD) has been reported.•The association of post direct-acting anti-viral agents with the development of both hepatocellular carcinoma (HCC) and CD may have immune base.•A new radiologic finding of hepatic CD in the form of vessels traversing through the lesion with no attenuation or occlusion has been encountered in this case.•Pathology remains the gold standard in the diagnosis of CD. CDCastleman diseaseUCDUnicenteric CDMCDMulticentricHVHyaline vascularPCPlasma cellHCCHepatocellular carcinomaDAAsDirect-acting antiviral agentsHCVHepatitis C virusSVRSustained virologic responseCTComputed tomographyGCGerminal centerFDCsFollicular dendritic cellsEGFREpidermal growth factor receptorRSReed Sternberg1Castleman disease (CD) is a lymphoproliferative disease reported first in 1954 as a localized lymph node swelling ,3. EightThe work has been reported in line with the SCARE 2018 criteria .2A 62 years old male, with no clinical relevant, medical, surgical or family history, received DAAs for chronic hepatitis C virus (HCV) related liver cirrhosis. Three months later, the patient achieved a sustained virologic response (SVR) and stuck to the HCC screening program. Unfortunately, the patient developed three hepatic focal lesions six months after the end of treatment. The patient referred to the oncology unit  for further evaluation. Laboratory tests: AST/ALT was 85/46 U/L, total bilirubin/direct bilirubin was 1/0.3 mg/dl, albumin was 3.8 g/dl, Hb was 10.6 g/dl, CRP was 2.8 mg/L, and INR was 1.43. Serum tumor markers were of average values; AFP was 1.7 ng/mL, CEA was 4.1 ng/mL, and CA19.9 was 15 U/mL. In the meantime, the patient underwent triphasic computed tomography (CT) scans of the abdomen and pelvis and revealed a cirrhotic liver with segment IV two hepatic focal lesions. Each measured 1 cm on the greatest dimension displaying typical criteria for HCC. Meanwhile, another large segment II mass measuring 5 cm on size showed a non-enhancing pattern throughout all phases, Postoperative surgical resection revealed a whitish, circumscribed, fleshy mass arising on top of well-established liver cirrhosis. Microscopic examination demonstrated a fairly circumscribed mass formed entirely of lymphocytes arranged in a nodular pattern. The nodular pattern formed of irregular hyperplastic lymphoid follicles of variable sizes surrounding by concentric rings of small lymphocytes represented expanding mantle zones “onion skin”, 3Castleman's disease is a primary lymphoproliferative disease affecting lymph nodes with rare reported cases in the extra-nodal site . CD is cEighteen cases of hepatic CD have been reported in the literature, including the present one. Most cases showed a female predominance 14/18) with a median age of 48 years old. Only a case was reported in a pediatric patient . The pre with a mThe pathogenesis of CD is not clear with several viruses that have been implicated in the pathogenesis of nodal CD ,10. A deThere were no specific radiological features have been proposed for hepatic CD. Contrast-enhanced CT showed a hypervascular pattern in ten cases, hypovascular in one case, and non-enhancing pattern in another case. The persistent enhancement pattern during the arterial and venous phases could be attributed to the narrowing of the arterial lumen and thickened fibrotic wall ,14. BesiSurgical resection and pathological evaluation remain the diagnostic method in the assessment of hepatic CD. Hepatic resection was performed in 16 out of 18 cases as a diagnostic and therapeutic tool. In the meantime, steroid therapy was done in two pathological approved CD cases ,16. The 4Despite the hepatic CD is a rare entity; we should put it in the differential diagnosis of a hepatic focal lesion with atypical radiological findings. The abstinence of characteristic clinical and radiological features makes pathological evaluation is the gold standard in hepatic CD diagnosis. The role of DAAs in the prevention or progression of hepatic CD necessitates further studies.Not commissioned, externally peer reviewed.Not required, all was done on the seek of diagnosis and proper management of the patient.No funding.All authors of this paper have participated in its drafting and approved the final version submitted. DS, HA, YF, and SK wrote the case report. TH, NE, DE, and DS contributed in the pathological diagnosis. YF contributed in the surgical part. HA provided the radiological imaging and interpretation of data. SK shared in the oncology section. AM performed the technical immunohistochemical work. MS and DS interpreted the immunohistochemical work. DS, NE, TH revised, and edited the final version.1.Name of the registry: Not applied.2.Unique Identifying number or registration ID:3.Hyperlink to your specific registration (must be publicly accessible and will be checked):Dina Sweed.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.All authors have no conflicts of interest to declare.


Pleomorphic xanthoastrocytomas (PXA) are rare, typically benign, slow-growing tumors that commonly occur in the cerebral hemispheres. We describe two cases of clinically aggressive PXA with uncommon locations; one was in the tectal plate, and the other had simultaneous multicentric lesions.The both cases presented with severe headache with no significant past medical history.PXA World Health Organization grade II were histopathologically diagnosed from surgically resected specimens, and immunohistochemical and sequence analysis revealed a high Ki-67 proliferative index and BRAF V600E mutation in both the cases.The first case presented with multicentric lesions and underwent partial resection, whereas the second case presented with a tectal plate tumor that was managed by gross total surgical resection. Strong 5-aminolevulinic acid (5-ALA)-induced fluorescence was observed in both the lesions. Postoperative radiotherapy plus concomitant and adjuvant temozolomide was administered to both the patients.Despite completing adjuvant chemo-radiotherapy, both the patients had local tumor recurrence at 2 and 5 months after the operation, respectively.The progressive clinical courses in our cases suggest that additional postoperative therapy should be considered during the treatment of PXA with a high Ki67 index, and that temozolomide with radiotherapy, followed by temozolomide maintenance therapy, may not prevent recurrence in such tumors. Importantly, our experience implies that unlike other subtypes of low grade gliomas, 5-ALA fluorescence is useful for intraoperative visualization of PXA. In contrast, the 5-year recurrence-free survival rate is significantly shorter (48.9%) in PXAs with anaplastic features; this type of lesion is now classified as anaplastic PXA (Grade III) in the 2016 World Health Organization (WHO) classification of Tumors of the Central Nervous System. Anaplastic PXA is defined as PXA with a mitotic index ≥5 per 10 high-power fields and/or necrosis. Although proliferative activity does not affect tumor grading in PXA, some authors have emphasized the use of the Ki-67 index as a prognostic factor.,7 Here, we describe two cases of clinically uncommon PXA with aggressive clinical courses, both with a high Ki67 index, wherein one patient presented with a multicentric lesion and the other, a tectal plate tumor.Very few reports have described simultaneous multicentric PXA.22.118F-fluorodeoxyglucose (FDG) uptake in both the lesions  and adjuvant  temozolomide following surgery. The reduction in size of residual tumor was not observed by adjuvant therapy. She remained stable without deterioration in her clinical symptoms, however, MRI at 3 months after the operation revealed tumor regrowth  revealed a distinct and enhanced mass lesion in the medial temporal lobe and the right orbital gyrus, both of which showed slight growth on a follow-up MRI at 1-month after initial presentation  and adjuvant  temozolomide following the surgery, despite which tumor recurrence was detected on follow-up MRI at 5 months after the operation , and is rarely seen in pilocytic astrocytomas and gangliogliomas.4We describe 2 cases of PXA with unusual clinical presentations and the case reports suggest that high Ki-67 LI in PXA can be used as a predictive marker of an adverse clinical course and that a combination of radiotherapy and chemotherapy with temozolomide may not be beneficial for patients with PXA with high Ki-67 LI values. Conversely, 5-ALA fluorescence is potentially beneficial for intraoperative tumor visualization in PXA.We wish to thank Kyoko Tsujikado  for providing technical support and Enago for the English language review. We declare that we have no conflict of interest.Conceptualization: Issei Takano.Data curation: Yoshiko Fujii, Yuki Inoue, Yoshiki Sugiura.Supervision: Ryuta Nakae, Kensuke Suzuki.Visualization: Yoshihiro Tanaka.Writing – original draft: Masaya Nagaishi.


The safe adoption of transanal total mesorectal excision (taTME) has occurred in Australasia as previously reported by the current authors. Planes beyond TME can be utilised in more advanced cases to achieve negative margins during transanal dissection.In this article we describe how taTME is used to perform an en-bloc partial vaginectomy and aid restore intestinal and vaginal continuity in a young female with a locally advanced rectal cancer and posterior vaginal wall involvement in the pre-treatment magnetic resonance imaging.The transanal technique allowed the surgeons to remove a disc of vagina, ensure organ preservation and control the main R1 risk point. An R0 resection was achieved.This technical note highlights that in experienced hands, taTME may be safely implemented to maintain restorative options in locally advanced rectal cancer requiring resection beyond the total mesorectal excision plane. MagneThe safe adoption of transanal total mesorectal excision (taTME) has occurred in Australasia as highlighted by the our group , and disThis case depicts the application of a taTME approach to perform an en-bloc partial vaginectomy with preservation of the cervix and uterus and restore intestinal continuity in a young female. Whilst the abdominal intent was to perform a robotic resection, despite an unexpected abdominal conversion the utility of the transanal technique is highlighted in this case.This work has been reported in line with the SCARE criteria  and appr1.12, presented with a locally advanced, mismatch proficient rectal cancer. She had had 12 months of symptoms, including altered bowel habit, rectal bleeding and increasing obstructive-type symptoms. Her initial staging with MRI and chest-abdomen-pelvis computed tomography (CT) showed a bulky mucinous rectal cancer with invasion of the posterior vaginal wall and possibly the cervix -positron emission tomography (PET)-CT and MRI. An exam under anaesthesia was also performed to assess vaginal involvement. These results confirmed an anteriorly based tumour, 6 cm from the anal verge with posterior vaginal wall invasion and potential cervical invasion, enlarged right pelvic side-wall lymph nodes and no further distant disease.A discussion at the exenterative multidisciplinary team meeting suggested utilising a robotic approach from above and taTME approach from below as a combined two-team approach.22.1The operation was carried out seven weeks after completion of chemoradiation. The operating team was set up for a synchronous two teams approach (Cecil approach) (surgeons SW/JM). The intent was to perform a robotic abdominal component including complete splenic flexure mobilisation being performed at the time of initial transanal set up. Unfortunately, at the outset of the robotic operating component a very proximal inferior mesenteric artery (IMA) injury occurred that required immediate but controlled conversion (only second reactive conversion in over 250 robotic cases). IMA bleeding was controlled using a gauze and a laparoscopic grasper to occlude the defect through the assistant port whilst the robot was de-docked and a midline laparotomy was performed. The abdominal surgery was continued as an open procedure. This included proximal suture of the IMA with interrupted 4-0 prolene, controlling the bleeding. The IMA was divided and suture ligated with 0 Vicryl and afterwards the left colon and sigmoid colon were mobilised in a lateral to medial fashion with identification and protection of left ureter, nerves and gonadals. Medially the peritoneum at the level of the sacral promontory was incised and continued cranially to the root of the IMA. Further dissection was carried out cranially to the duodenal-jejunal ligament allowing proximal ligation and division of the inferior mesenteric vein. Complete splenic flexure mobilisation was performed following access into the lesser sac via division of supra-colic omentum with continuation laterally to meet the lateral dissection plane along Toldt´s fascia. The colon was transected proximal to the level of the IMA with 2-0 Vicryl ties to the mesenteric vessels and marginal artery and division of the colon performed with a GIA™ 80 mm linear stapler. The abdominal contents were packed superiorly to allow commencement of the abdominopelvic dissection.The abdominopelvic approach included right ureterolysis, posterior TME plane dissection followed by dissection of the superior pelvic peritoneum in the pouch of Douglas to facilitate partial mobilisation of the recto-vaginal septum anteriorly and the entry into the posterior vagina. Despite preoperative concerns about posterior cervical invasion, this was clearly not the case intraoperatively where planes were easily dissected and non-involved up to the cranial portion of the posterior vagina.This component was then ceased until the taTME team developed the anterior plane between rectum and vagina in order to control the potential R1 resection margin. The transanal component defined the distal vaginal entry point.A complete right lateral pelvic side-wall dissection was performed commencing at the level of the common iliac bifurcation dissecting inferiorly into obturator fossa, preserving the obturator nerve and vessels. Medialisation of the right ureter was necessary. The nodal harvest of obturator fossa and the internal iliac system was performed. A full description of our approach to a side-wall has been provided previously .2.2The taTME setup was performed synchronously. A Lone star retractor  was applied allowing eversion of the anoderm. The GelPOINT® Path  was placed in the anal canal and thorough washing with cetrimide performed. A gauze was placed in the anus, and 1-0 prolene suture with a 26 mm rounded needle was used to create the pursestring in a standard clockwise fashion ensuring equidistant sutures from the port cuff a. Three The specimen was extracted transabdominally with primary repair of the posterior vaginal wall with interrupted full thickness 2-0 Vicryl sutures. A colonic pouch was fashioned, and a single stapled anastomosis created with an EEA™ 33 mm circular stapler (Medtronic).3Total operative time was 220 min and blood loss approximately 400 mL. The patient had an uneventful recovery and was discharged on day six post-operatively.3.1On macroscopic inspection of the specimen inspection the vaginal disc looked complete with clear margins and the mesorectum was intact throughout . Histopa3.2The patient completed adjuvant chemotherapy (CAPEOX), and had her stoma closed 10 months after surgery. After 12 months of follow-up she has no local or distant recurrence.4This case highlights the merits of adopting a combined abdominal and taTME approach for surgical resection of complex anterior rectal tumours to achieve an R0 resection. A partial posterior vaginectomy has been performed with preservation of the uterus utilising this approach and hence allowed for colonic and vaginal reconstruction. To our knowledge, this is the first reported case in the literature.Planes beyond the TME dissection can be utilised in select cases to allow for multivisceral resection ,8. Here,The case again highlights the importance of having specialist colorectal surgeons involved in the utilisation and adoption of new technology. Both SW/JM are exenterative minimally invasive surgeons and SW/AH are credentialled robotic and taTME proctors. The technique represents an incremental alteration as we move forward with more organ preserving approaches for complex rectal cancers. The personalised decision to preserve the uterus was made due to the patients young age and possible future fertility as the fertility field grows in the future, despite having had chemoradiotherapy. The intraoperative findings supported such an approach.The authors recommend that such approaches occur in institutions that are high volume rectal cancer centres with appropriate specialised experience.5TaTME can be safely implemented to maintain restorative options in locally advanced rectal cancer requiring resection beyond the mesorectal plane.The authors declare that they have no conflict of interest.Epworth Medical Foundation.This study is exempt from ethnical approval in our institution.Informed consent was obtained from all patients included in the study and a copy is available for the Editor-in-chief by request.Satish Warrier, Jacob McCormick, Alexander Heriot, Peadar Waters and Jose T. Larach were the surgeons involved in this case. Satish Warrier and Jacob McCormick were the primary surgeons. Jose T. Larach, Peadar Waters, Philip Smart and Satish Warrier wrote the article. Al authors reviewed the final version of the manuscript.NA.Satish K. Warrier.Not commissioned, externally peer-reviewed.


A seven-year-old female spayed Australian Shepherd was presented for a 3-day history of left eye ventromedial strabismus, episcleral injection, protrusion of the third eyelid, miosis, and enophthalmia. Magnetic Resonance Imaging (MRI) identified lesions in the left medial pterygoid muscle and left tongue base. Cytology and histopathology revealed pyogranulomatous inflammation with rod-shaped bacteria and pyogranulomatous myositis, respectively. One month of oral antibiotics resolved both lesions. Repeat MRI showed a mild decrease in size of the left medial pterygoid muscle consistent with fibrosis. Clinically, residual, positional ventral strabismus remained upon dorsal neck extension, but all other ophthalmic abnormalities resolved. To the authors' knowledge, this is the first report of pyogranulomatous myositis causing this constellation of clinical signs and of repeat imaging depicting resolution of these lesions with therapy. A seven-year-old, female spayed Australian Shepherd was presented for severe chemosis and episcleral hyperemia of the left eye (OS). The remainder of the examination was unremarkable. Three days later, the eye had ventromedial strabismus with a positive passive forced duction test, and anterior movement of the third eyelid was restricted when grasped with forceps . The eye*-weighted gradient recalled echo (GRE) images , and transverse T2-weighted TSE with Variable Flip Angle (“SPACE”) . Post-contrast  sequences included transverse T1-weighted Volume Interpolated GRE images with fat saturation (“VIBE”) , sagittal T1-weighted images , transverse T1-weighted images , and dorsal T1-weighted images with fat saturation .The dog was anesthetized and placed in dorsal recumbency for Magnetic Resonance Imaging (MRI) of the head using a 1.5 Tesla superconducting MRI system  equipped with a head and neck coil. Pre-contrast MRI sequences acquired included sagittal T2-weighted spin echo images , dorsal short tau inversion recovery (STIR) images , transverse T2-weighted images , transverse T1-weighted images , transverse T2-weighted fluid attenuated inversion recovery (T2-FLAIR) images , transverse proton density (PD) weighted images , transverse T2*-weighted images. Pinpoint intralesional T1 and T2 hypointense foci were noted. The lesions were strongly contrast enhancing and mildly heterogeneous in intensity. The left mandibular and medial retropharyngeal lymph nodes were mildly enlarged and heterogeneously contrast enhancing. No intracranial abnormalities were identified. Ultrasound guided tissue sampling or surgical biopsy of the lesion at the tongue base was recommended.Two separate lesions with remarkably similar imaging characteristics were associated with the soft tissues of the head. One was associated with the left medial pterygoid muscle immediately ventral to the orbital fissure . It measUltrasound examination of the ventral tongue was performed using a high frequency linear transducer . The tongue base lesion appeared well-circumscribed, hypoechoic, and fairly homogeneous .Cytology of a fine needle aspirate of the tongue base mass revealed pyogranulomatous inflammation and rod-shaped bacteria. An incisional biopsy of this lesion revealed pyogranulomatous myositis, possibly secondary to prior foreign body penetration. Foreign material was not identified within the submitted biopsy sample. A tissue culture was not done, but, in retrospect, should have been submitted. Sampling of the pterygoid muscle lesion could not be performed due to proximity to the maxillary artery.The patient was placed on oral amoxicillin/clavulanic acid  for 1 month and the clinical signs resolved. Ten weeks after presentation, the ophthalmic examination was normal except for residual, positional ventral strabismus OS during dorsal extension of the neck .A repeat MRI examination was performed 10 weeks after presentation. The protocol was identical to the initial study. The previously seen soft tissue lesions associated with the left medial pterygoid muscle and the tongue base were largely resolved. Faint regions of altered signal intensity  were still noted . The lefThis report describes a unique cause of ventral strabismus in a dog. Given the initial clinical signs of resting ventromedial strabismus, retrobulbar soft tissue pathology was suspected. Strabismus can be classified in many ways, such as resting vs. positional and restrictive vs. neuronal. A “resting” ventral strabismus is most indicative of a restrictive process, such as fibrosis or muscle impingement, that prevents proper relaxation of specific muscles. A “positional” strabismus is only seen in certain head orientations and most commonly occurs from a vestibular neuropathy . In the On presentation, the restricted globe movement, ventromedial strabismus, and miosis indicated a combination of a neuropathy of the branches of CN III and inflammation of the musculature. Once CN III exits the orbital fissure, several branches run through the orbit to innervate the parasympathetic iris and ciliary body muscles, the dorsal, medial, and ventral rectus muscles, and the ventral oblique muscle. The medial pterygoid muscle forms part of the posterior orbital floor . The intTwo intramuscular lesions were appreciated on MRI. Differential diagnoses for intramuscular nodules and masses in humans include primary and metastatic neoplasia, hematomas, nodular myositis, sarcoidosis, crystal deposition and post injection changes . DependeReports of imaging findings in dogs with muscle lesions are limited. Inflammatory myopathies encompass a large group of heterogeneous disease processes of various etiologies including infectious and immune-mediated etiologies. Even though reported MRI features are variable, these conditions often cause multifocal patchy, ill-defined intramuscular lesions rather than distinct nodules as seen in our case –7. AbnorUpon questioning, the dog's owners mentioned that the dog frequently chews on sticks, but they had not noticed any clinical signs commonly seen with acute penetrating oropharyngeal injuries such as retching, salivation, or pain. The pathology results in our patient suggested a prior penetrating injury; however, it is worth noting that foreign material was not identified on imaging or histopathology. It is conceivable that the ends of a sharp-edged stick penetrated both the dorsal and ventral lining of the oral cavity, introduced bacteria into the soft tissues, and caused the inflammatory lesions found on imaging without actually leaving foreign material behind. Alternatively, it is possible that foreign particles were left behind but were too small to be seen on imaging or histopathology. Little is known about the accuracy of MRI in detecting foreign bodies in soft tissues. In one experimental study, acute wooden foreign material appeared T1 and T2 hypointense to surrounding musculature . Even thMetastatic disease to muscle may occur secondary to sarcomas , carcinomas , and round cell tumors . Most reports of imaging findings with muscle metastases in dogs are based on whole body computed tomography studies , 13. On A repeat MRI was done to investigate if the resolution of clinical signs corresponded to resolution of the lesions noted on the initial MRI. The decrease in muscle volume and mild diffuse signal intensity changes seen associated with the left medial pterygoid muscle are consistent with fibrosis and correspond to abnormalities reported with other fibrotic myopathies in dogs , 7, 15. In conclusion, this report describes the MRI findings of a dog with pyogranulomatous myositis of the tongue base and left medial pterygoid muscle resulting in ventromedial strabismus OS. Rather than being ill-defined, these inflammatory lesions were distinct nodules. The current patient had a history of gnawing on various objects, including sticks. Therefore, both lesions could have occurred from an object penetrating the tongue base and retrobulbar space; however, no foreign material was found on histopathology and no traumatic event was observed. While no culture of the tongue base lesion was performed, cytology and histopathology revealed rod-shaped bacteria and myositis, respectively. Anaerobic and aerobic cultures should be performed for similar appearing lesions to help with diagnosis and guide antibiotic therapy. Given their appearance, both lesions were assumed to be from the same process. A one-month course of a broad-spectrum antibiotic resolved the lesions confirming this assumption. A repeat MRI examination showed absence of retrobulbar inflammation and fibrosis of the left medial pterygoid muscle correlating with resolution of previously noted neuro-ophthalmic abnormalities and only persistent, clinically evident positional ventral strabismus, respectively.All datasets presented in this study are included in the article/supplementary material.Ethical review and approval was not required for the animal study because the report was written retrospectively on a patient treated with standards of care at a veterinary teaching hospital. Patient care, including diagnosis and treatment, did not include methods intended for research. Written informed consent for participation was not obtained from the owners because ethical approval and written consent from the owner were not needed for this report. Oral consent was given by the owner.Conception and design done by AS, DH, and SH. Acquisition of data done by all authors. Drafting of the article as well as article revision done by all authors. Final approval of the completed article done by all authors.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.


Pituitary stalk interruption syndrome (PSIS) is an extremely rare cause of growth failure and delayed puberty. It can be diagnosed by magnetic resonance imaging (MRI) of the hypothalamus and pituitary gland, showing an ectopic or absent posterior pituitary, an absent or interrupted pituitary stalk, or small anterior pituitary, in combination with growth hormone or other pituitary hormone deficiencies. The exact etiology of PSIS is unknown. In this article, we describe two cases of PSIS in Syria which are, as far as we know, the first published cases. Growth failure is one of the most common reasons for referral to the endocrine clinic. Although the most frequent causes of short stature are constitutional and genetic, endocrine disorders, especially growth hormone (GH) deficiency, should always be considered . PituitaIn this article, we describe two cases of PSIS that were diagnosed in boys at different ages from Syria.A 7-year-old boy was referred to the endocrine clinic for short stature. He was born at full term without complications. His birth weight was 3 kg, and his family history was unremarkable. During childhood, he had all his vaccinations and had not been hospitalized.2). Compared to his midparental predicted length (119 cm), he was 3.6 standard deviation (SD) points below expected. Heart and breath sounds were normal and so was the abdominal examination. Testicular volume was 3 ml each, measured by using a Prader orchidometer. Pubic hair distribution and penile size (3 by 1 cm) were both consistent with Tanner stage 1.On examination, he seemed well without any abnormal facial features, and he was 19 kg in weight and 101 cm in height (body mass index (BMI) 18.62 kg/mμcg of clonidine was consistent with GH deficiency. Other pituitary hormones were also evaluated and revealed central hypothyroidism and adrenal insufficiency . Compared to his midparental predicted length (169 cm), he was 3 SD points below expected. Auscultation of the heart and lungs did not reveal any abnormalities. Abdominal exam was normal. Testicular volume was 4 ml each, measured by using a Prader orchidometer. Pubic hair was consistent with Tanner stage 1. Penile size was 2 by 1 cm, consistent with micropenis. No gynecomastia was noted.He was born full term in a breech position without complications and had a birth weight of 2.3 kg. No hypoglycemia or other complications were reported during childhood. On examination, he had no abnormal facial features and looked well. He weighed 43 kg and was 150 cm in height  was initially described and defined by Fujisawa et al. in 1987 . PSIS isThe exact prevalence of this syndrome remains unknown. Less than 1000 cases were reported in the literature until 2010 . The ageBreech delivery, cesarean section, and neonatal hypoxemia were suggested as potential causes of this syndrome . HoweverWhile our second patient had a breech delivery, our first patient underwent uncomplicated vaginal delivery and had a negative family history. The presence of multiple hormone deficiency suggests a genetic etiology. However, due to lack of availability of genetic testing assays in our country, this was not performed.In most published series , 4, 8–13TSH deficiency was the most common deficit (79.5%) alongside GH deficiency in PSIS patients, and ACTH deficiency also was common (67.5%), followed by FSH/LH deficiency in 65.1% of patients .Our two cases had GH, TSH, and ACTH deficiency. LH/FSH deficiency may be detected in minipuberty or discovered later in peripuberty. Therefore, gonadal assessment should be conducted regularly in patients with PSIS . While hThe incidence of extrapituitary manifestations in PSIS is high . MidlineThe lifelong prognosis depends on the time of diagnosis and early treatment of hormonal deficiency .Close monitoring of a child's growth over time helps in early detection of growth failure and early treatment of the underlying causes. Rare etiologies such as PSIS should be kept in mind especially in multiple hormonal deficiencies, as it carries good long-term prognosis if diagnosed early on and treated adequately.Finally, these two cases are, as far as we know, the first published cases from Syria.