We present the case of a 67-year-old male patient with a past history of previously resected T3 right adrenocortical carcinoma and T3N1 signet ring cell adenocarcinoma of the stomach who presented with recurrence of gastric cancer in the form of a large solitary mass in the right abdomen. He was treated with ECX  chemotherapy and multivisceral resection. This recurrence pattern is the first such description in the literature, and we discuss the controversies and arguments in favour of offering surgical resection. Linitis plastica, also known as signet ring adenocarcinoma of the stomach, although rare but rising in incidence, is associated with a poor prognosis and high recurrence rates following surgical resection. It accounts for about 10% of all gastric cancers and is characterised by rigidity and fibrotic thickening of the stomach wall. Standard treatment pathways are those of gastric cancer, and surgical resection remains the only recognised therapeutic option offering a potential cure. Recurrence is often in the form of disseminated peritoneal disease.We report the case of a 67-year-old retired consultant gastroenterologist with a past medical history of primary hypothyroidism and past surgical history of a right adrenalectomy and nephrectomy with inferior vena cava (IVC) resection in 2008 for a pT3 high-grade adrenocortical carcinoma. Complete resection of the tumour was achieved microscopically with free-floating tumour extension into the intrahepatic inferior vena cava. The tumour was ruptured intraoperatively, and he was therefore advised to start long-term adjuvant mitotane. This caused chronic adrenal insufficiency, treated with replacement therapy, and significant hypercholesterolaemia. This is believed to have lead to a symptomatic superior mesenteric artery stenosis presenting in 2010, which was successfully treated with a superior mesenteric artery stent.In January 2012, he presented with melaena and was found to have a distal antral gastric carcinoma, infiltrating submucosally, biopsies of which were consistent with linitis plastica. He went on to have a subtotal gastrectomy, cholecystectomy, and D2 lymphadenectomy with neo-adjuvant and adjuvant ECF chemotherapy (3 + 2 cycles). The 5-FU was replaced by raltitrexed due to minor asymptomatic coronary disease and the sixth cycle was omitted as he moved to a different region of the country. Histopathology showed a poorly differentiated diffuse type adenocarcinoma of the antrum, staged as pT3 N1 (1/27) V0 R0, Mandard TRG 3. The mitotane was stopped at the start of chemotherapy as no recurrence of the adrenocortical carcinoma was evident on follow-up imaging. He remained well until August 2014 when he developed early satiety and underwent gastroscopy, which showed no macroscopic abnormality, and random biopsies from the staple line showed no malignancy. The patient presented again in October 2014 when he developed further abdominal pain. Computed tomography cross-axial imaging showed a 14 cm mass around the ascending colon and hepatic flexure with a suspicion of retroperitoneal and psoas involvement, as illustrated in Figure A tunnelled transcolonic biopsy of this mass revealed the recurrence of the gastric adenocarcinoma. Further imaging in the form of a PET scan in December 2014 showed moderately FDG-avid (maximum SUV 9.2) wall thickening of the large bowel at the hepatic flexure extending over a length of 10 cm, abutting the right psoas muscle; the uptake in the muscle was thought to be physiological.The case was discussed at the referring hospital's multi-disciplinary team meeting (MDT), and he was offered six courses of palliative EOX chemotherapy. At the patient’s request, he was also referred to our institution for a second opinion. He was subsequently seen in the oncology and surgical clinics and discussed a number of times at our regional MDT. On review of further imaging from March 2015, it was felt that the edge of the mass was not infiltrating the psoas fascia and that the retroperitoneal fascia was intact, taking account of the postoperative changes from his previous adrenal surgery. It was also noted that there was encasement around the pancreas and focal dilatation of the distal common bile duct due to a mesenteric invasion, although there was no significant derangement in liver function tests. Following extensive discussion, the final suggestion was for the patient to be offered surgical resection after completing six cycles of EOX . Informed patient consent was obtained. No identifying patient information is disclosed in this paper. On July 22, 2015, the patient underwent a right hemicolectomy with en bloc pancreaticoduodenectomy, IVC resection, and retroperitoneal lymphadenectomy. The histology reported that sections showed the presence of fibrosis in the wall of the colon and in areas of adhesion between the large and small bowel. Within the fibrous area, there was an uneven infiltrate of poorly differentiated adenocarcinoma with diffuse/signet ring cell morphology. The carcinoma was present in the submucosa and muscularis propria of the colon as well as the surrounding fibrous tissue of the adhesion, but there was no definite lymphatic or venous invasion. No tumour was seen in the four lymph nodes identified and the area of infiltration appeared completely excised. Immunohistochemistry highlighted that the tumour also extended into the colonic mucosa, and was negative for HER2. The patient made an uneventful recovery, interrupted only by admission with an intra-abdominal collection, successfully treated with percutaneous drainage. Follow-up CT in November 2015 showed no evidence of disease recurrence.Recurrence from gastric linitis plastica tumours is often in the form of the disseminated peritoneal disease and standard treatment pathways are those of gastric adenocarcinoma. However, there is increasing evidence that linitis plastica tumours of the stomach should be considered a different disease entity from non-linitis plastica adenocarcinoma . FurtherIt is clear that the timing and location of the lesion would have been more likely to represent a recurrence of the adrenocortical carcinoma, and this highlights the importance of histological confirmation. There are, moreover, several atypical features of this pattern of linitis plastica recurrence that should be highlighted. As mentioned, review of earlier imaging suggested that there might have been an area of thickening in the wall of the colon at the hepatic flexure prior to the detection of the mass. This suggests that the recurrent lesion may have started in the colon rather than the retroperitoneum (as supported by the postoperative histology). Secondly, the presence of a large mass after only eight months without any obvious other sites of disease is not a characteristic pattern of recurrence for linitis plastica tumours. For haematogenous metastases of linitis plastica, there is no proven role for surgery, and this was the justification for the treatment recommendation from the referring hospital, which in itself appears evidence-based. In this case, however, there was no evident disease in more proximal sites for the haematogenous spread, such as the liver or lung, raising the question of whether this represented a local recurrence. The fact that the recurrence was solitary and had remained solitary for a reasonable period of time, in addition to the atypical location for this histological tumour type, were significant factors contributing to the decision to offer surgical resection. A final interesting consideration in this case, although less likely, is whether this tumour could represent a third primary, as primary linitis plastica of the colon has been reported . The patThis specific recurrence pattern represents, to our knowledge, the first such case described in the literature. The fact that surgical clearance required a major multivisceral resection with significant potential morbidity, coupled with the uncertain efficacy of palliative chemotherapy, highlights the dilemma faced by clinicians in advising the most appropriate treatment strategy. All such cases should be discussed in a specialist MDT and any surgical intervention should be reserved for patients who are fully informed of the available evidence base, morbidity profile associated with the surgery, and rationale for such treatment.


This paper presents a review of a dog, with a retrobulbar chondrosarcoma, which was admitted for surgery for visible changes in his eye during inspection. Orbital neoplasia in dogs may be primary and secondary. Sixty percent of orbital neoplasia in dogs are primary, ninety percent of which are malignant. Retrobulbar neoplasms are rare and in their early stage represent a diagnostic challenge. Chondrosarcoma of the skull is a slow-progressing malignant disease which occurs locally, aggressive with invasion into the surrounding tissues. Dogs with chondrosarcoma of the skull have life expectancy between 210 and 580 days - in our case it was 180 days - after the first alterations on the eye of the dog occurred. Chondrosarcoma (CS) is a malignant tumour of bones in animals and humans. This neoplasm corresponds to 5 - 10% of all the primary bone tumours in dogs. Primary CS may occur on skeletal system and then it is called central or medular CS. When CS is located on periosteum, it is called peripheral CS .Eight years and seven months old male Cocker Spaniel was admitted to the surgery for presence of a thick mucus discharge and changes on the cornea of his left eye. The dog apparently did not have any other health issues. During the clinical examination, the dog was fit, in a good mood and there were no changes in regional lymph nodes. The changes on the left eye were visible: presence of the dried mucus discharge and clingy hair on the upper eyelid and medial canthus.Due to protrusion of the eye bulb, a pronounced exophthalmos of the left eye was clearly perceptible. The upper lateral quadrant of the cornea was blurry. There were no visible changes in the right eye.The methods used during the first examination of the eye were: the fluorescein eye stain test, the Schirmer I tear test, measurement of the intraocular pressure and the Jones’ test for patency of nasolacrimal duct. As the process progressed, the tumour was sampled for histopathological examination and computed tomography of the skull was also performed in the follow-up examinations.The intraocular pressure was measured by the Schiotz method, with prior application of anaesthetic (proparacain) to the eye. The solution of fluorescein was used for staining the cornea and testing the patency of the nasolacrimal duct. The computed tomography of the skull was performed by the third-generation device GE (9800-8800), under sedation using Domitor.The inspection revealed changes on the auxiliary parts of the left eye and presence of dried mucus discharge. Alopecia was not noticed on the lids of the left eye. The third eyelid protruded out of its place covering half of the eye bulb .The dog did not react painfully to retropulsion of the eye bulb, which was performed due to its enlargement. There was no counter effect from the bottom of the orbit of the affected eye during the retropulsion. The Schirmer I test results for the right and the left eye were 15 mm and 6 mm, respectively. There was a superficial whitish blur in the upper medial quadrant of the left eye cornea.The fluorescein stain test was positive, which indicated alteration of the superficial layer of the cornea. The Jones’ test was positive for both nasolacrimal ducts. The changes on the iris and on the anterior chamber were not noticed during the ophthalmoscopy. Examination of the posterior segment of the eye and the lens of the left eye was performed after application of the Midriacil 1% drops to the eye. A central Y nuclear sclerosis was noticed in the lens.There were no anatomic deviations on the fundus, both on the tapetal and non-tapetal part of the retina. A slight swelling of the optic papilla was observable. The mean values of the intraocular pressure of the right eye and the left eye were 22.38 mmHg and 10.24 mmHg, respectively.The findings of the examination, absence of tears, moderate exophthalmos and slight protrusion of the third eye lid were basis for establishing the working diagnosis of keratoconjunctivitis sicca of the left eye, so further regular follow-ups were advised. The therapy for dry eye  and systemic antibiotic therapy  were administered to the dog.A month later, the next examination revealed significant deterioration of the clinical signs on the surface of the cornea and significant enlargement of the eye bulb .The position of the left eye bulb indicated upward lateral strabismus. The third eye lid was everted and the conjunctiva was swollen. When performed again, the Jones’ test resulted with absence of staining in the left nostril which indicated obstruction of the left nasolacrimal duct.The owner reported that the dog sniffles discharging a thick secretion from the left nostril and has swallowing problems. Computed tomography of the skull with prior application of sedative Domitor followed the examination of the mouth cavity, which revealed an obvious tumoural mass behind the last upper molar on the left side.The scan was performed in dorsolateral position .The images clearly showed presence of the neoplasia in the region of the medial orbit, osteolysis of the nasal septum and presence of the tumoural mass in the left nasal duct  and 5.The excisional biopsy of the neoplasm in the mouth cavity with prior application of xylocain 2% spray was performed after the scan .The system therapy including antibiotic and NSAID – antibiotic Cephalexin at a dose of 15 mg/kg p/o and Movalis tablets at a dose of 1.7 mg/kg BW per day – was applied, as the dog obviously suffered from pain. The excised tumour was assessed morpho-pathologically.The tumoral mass was 3, 4×2, 5×1.8 cm in size and 25g in weight and hard in texture .The preparation for histopathological examination was dyed with hematoxylin and eosin (H&E). Histopathological findings showed presence of pleomorphic cells with vesicular nucleus containing one or more nucleoli. The tumour cells produced the chondroid matrix and they were arranged, by their shape and size, in irregular groups which were separated from the surrounding tissue .Such histopathological finding indicated a highly differentiated mesenchymal sarcoma of the left eye with destruction of the nasal septum.Since surgery and radical removal of the neoplasm, as well as application of chemotherapy, were not possible, the dog was given the medication for pain relief and the eye was treated locally with the antibiotic drops (Tobramicin 4×1). Three months after the final diagnosis was established, the dog died.Primary neoplasia of the orbit can arise from any tissue: epithelial, vascular, nervous or connective tissue. Secondary orbital neoplasia arises from the nasal structures, sinuses or cranial cavities of the skull and rarely as distant metastasis. Meningioma, lymphosarcoma, adenocarcinoma, fibrosarcoma, myxoma, glioma, squamous cell carcinoma (SCC) and rabdomyosarcoma are among the numerous neoplasia of the orbit in dogs.et al., 2007).In dogs, 90% of orbital tumors are malignant. CS occurs on pelvis, facial bones, ribs, vertebrae and scapula as a non-painful tumoural mass . The occurrence in trachea, lungs, omentum, mammary gland, heart valves, aorta, tongue, root of the penis and the penile urethra was also reported .CS is a malignant tumour cells which produce cartilaginous matrix. It develops from the cartilage structures of the bones and can also develop out of the tissues where cartilage normally does not exist. CS of the skull rarely occurs and most often, in 28.8% cases, it can be found in the nasal cavity, 9% of which is on the facial bones  , plain radiography of the skull, computed tomography and magnetic resonance imaging (MRI) are also the methods used in diagnosing CS of the orbit. Regarding the differential diagnosis, CS should be distinguished from non-neoplastic inflammatory orbital granuloma, cellulitis and abscesses and from myositis of extraocular muscles and masticatory muscles.The main symptom in the dogs with inflammatory processes in the region of the orbit is presence of a strong pain during mouth opening. The dogs with inflammatory processes in the region of the orbit feel pain during mouth opening .The following six parameters are being used in CT scan and in diagnosis of tumours of the nose, the superior sinuses and the orbit:Bilateral expansion of the tumour in basal and paranasal sinuses.Destruction of the nasal bone and finding of tumoural mass on nasal surface and in the facial area.Presence of the tumour in the mouth cavity with hard palate destruction.Presence of the tumour in the orbit and lateral pressure on the bulb.The frontal sinus involvement.et al., 2008).The brain involvement and destruction of the cranium, expansion of the tumour into the brain . All degrees of differentiation occur; benign tumours have an abundant cartilaginous matrix whilst malignant tumours contain closely packed cells with plump, frequently multiple nuclei. Mitotic figures are common in the more malignant tumours .Sometimes, calcifications and ossifications can be found in the cartilaginous tissue of the tumour. Treatment for CS of the skull is directly dependent on its localization and the degree of invasiveness towards the surrounding tissue. As it is a malignant tumour, the treatment of choice, if it is possible, would be the surgery.et al., 2008). In the case of the surgery, the operation is difficult because differentiation between the normal and the tumoural tissue is not easy.Orbitotomy with resection of zygomatic arch is recommended. If the tumoural mass is small, a transconjuctival approach to the tumour is also possible . In the case that the tumour invaded the brain, the mouth cavity, or the orbit, radiotherapy – either as monotherapy or combined with chemotherapy – and sometimes (as it was in our case) even only the antibiotic and NSAID treatment are recommended before the surgical treatment.Prognosis for the orbital tumours is poor since the surgical treatment does not solve the issue of recurrences. The dogs with orbital osteolysis (as in our case) have even poorer prognosis. In the case of radical surgery (orbitectomy), the owners are reluctant to accept the fact that their pet is going to have the postoperative facial deformity. The studies suggest that surgical treatment of CS and chemotherapy can prolong life of the dog for six months . The average life expectancy for dogs with CS that also invaded the nasal cavity is between 210 and 580 days, with treatment provided (rhinotomy with radiotherapy) . The low-grade chodrosarcoma can grow for a long time during several years without showing a tendency to metastasize, while the fast-growing CS metastasizes to lungs, kidneys, liver and heart .CS of the orbit is a rare, slow-progressing malignant disease in dogs and is extremely destructive to surrounding tissues. In the cases of clinically diagnosed CS in dogs, the dogs were in the age of about eight years. On the computed tomography scans one can observe a large, irregularly shaped mass with destruction of the orbital bones and mineralisation of the tumoural matrix. There is also compression of the local tissue, the muscles of the eye bulb and dislocation of the eye bulb from the orbit.Due to the difficult approach to the tumour and the pronounced destruction of the surrounding tissues, it is difficult to perform the surgery. Recurrences are very frequent and the life expectancy in dogs is less than a year after the accurate diagnosis is established. Due to the severe pain, it is necessary to apply the potent NSAID and opioid analgesics as well as the supportive therapy including vitamins and intravenous solutions to bring some relief to the dog.


Until now, less than ten cases of extra-adrenal chromaffin cell tumors have been reported to be localized to the spermatic cord area. All published studies report benign tumors with a diameter <2–3 cm and no invasion of the testis. In this article, we present one case of a giant malignant paraganglioma in the testis of a patient who had initially been operated for a giant mass in the scrotum. The mass developed in approximately 4 months. This is the first study reporting the following findings: i) paraganglioma was found exclusively in the testis, invading the testicle and not the spermatic cord, ii) it was malignant with lung metastasis, and iii) its size was 17.5 cm×10 cm×9.5 cm. We present the first – giant – malignant paraganglioma. Moreover, it is the first case report of a paraganglioma in the testis.This is the first study reporting the following findings:Paraganglioma found exclusively in the testis, invading the testicle and not the spermatic cord.It is malignant with lung metastasis.It is of the size 17.5 cm×10 cm×9.5 cm. The detection of paragangliomas has been reported to be extremely rare in the literature. Initially, these masses of chromaffin cells originating from the neural crest had been introduced as extra-adrenal pheochromocytomas, due to their catecholamine secretory capability; however, paraganglioma has become the commonly accepted term in the past decade. The most common sites of these tumors are the abdomen, the retroperitoneum along the para-aortic axis, the thorax, the cervical area, and the head. The testis and the spermatic cord are very rare locations. The first report of this mass was documented in 1971 by Eusebi & Massarelli The patient, a 66-year-old man, was admitted with a 4-month history of a gradual increase, in size of a, non-tender, mobile, big mass of the right hemiscrotal sac, following heavy weight lifting. The mass had been causing mobility problems to the patient due to its great size. There were no history of urinary symptoms or trauma of the scrotum.The first impression following the initial clinical examination was that of a non-complicated giant inguinoscrotal hernia. The left testicle was normal. There were no palpable inguinal lymph nodes. The routine hematological and biochemical markers were normal. He had a history of high blood pressure, psoriasis, and hepatitis B and C with two previous operations . His family history was unremarkable.The patient was operated for exploration of his right inguinal canal and the scrotal sac. There were no signs of hernia and the identification of the right testicle was not feasible, due to its encapsulation in a slid mass that was arising from the cord. The contents of the spermatic cord were ligated and cut. The lump was excised en block with very little blood loss and no significant hemodynamic changes under general anesthesia. There were no pre- or post-operative complications from surgical and anesthetic points of view. The patient was discharged 3 days after the operation. During his hospitalization, there were no disturbances in his blood pressure.The histological examination following the excision showed the presence of a neuroendocrine tumor of size 17.5 cm×10 cm×9.5 cm . The tumCD56 (NCAM1), patchy intense positivity for synaptophysin (PLAP (PLAA), CD30 (TNFRSF8), CK8/18 (KRT18P18), CK7 (KRT7), CK20 (KRT20), desmin, myogenin, and chromogranin. S100 protein revealed the presence of spindle cells at the periphery of the neoplastic nests, similar to the sustentacular cell pattern of a paraganglioma.The tumor shows a nested architectural growth pattern with expanded vascular spaces between the neoplastic nests, resulting in macrotrabecular morphology. Focal and confluent necroses are encountered. Neoplastic cells are medium to large in size, partially polygonal with plenty of eosinophilic cytoplasm. The nucleus is usually oval shaped and moderately pleomorphic, occasionally with prominent nucleoli. Additionally, multinucleated and bizarre cells could be observed, as well as many mitoses up to 12/10 HPF. In addition, plenty of vascular neoplastic emboli  in the ttophysin  and negaAfter the diagnosis of the paraganglioma, the patient was readmitted to the hospital for further investigations. Plasma and serum levels of epinephrine and norepinephrine were in the normal range and the tumor markers negative. The 24 h urine collection showed normal levels of vanillylmandelic acid. The ultrasound scan of the adrenal glands was unremarkable. The patient also underwent computed tomography of the brain, cervix, thorax, abdomen, and pelvis. The scan showed the following features:Multiple nodular lesions in both lungs with the larger one of diameter 4.1 cm  and locaNodular lesion left subdiaphragmatically=2 cm (possible lymph node).No inguinal lymph nodes.No fractures.No sign of primary lesion in the adrenal glands or any other sites.Owing to the absence of any lesion in the adrenal glands or any other sites toward the neural crest, primary paraganglioma was diagnosed in the right testicle. The patient was then referred to the oncologists for further treatment, however he refused any further treatment or intervention and passed away three months later due to myocardial infarction.Based on our literature search, this is the first reported paraganglioma sited exclusively in the testis and not in the spermatic cord where the vast majority of the paratesticular tumors are found, giving birth in this way to a new subcategory of testicular tumors The authors of previous case reports have supported the finding that the small size of the paragangliomas in their cases along with the consequent low ability to secrete high quantities of catecholamines was responsible for the absence of significant hemodynamic symptoms.In this case, although the size of the tumor was much bigger than all the other reported paratesticular paragangliomas, the absence of important hemodynamic variances along with the normal levels of the catecholamines in the serum and the urine is remarkable. Maybe the extra-adrenal chromaffin cell tumors located especially in the paratesticular areas as well as those in the testis lack the ability to secrete high amounts of catecholamines regardless of their size. However, the distant metastasis is a finding that can be related to the large size of the tumor in our case and indicates that early detection and excision of these tumors can be critical for the proper management and the survival of these patients.The patient's consent could not be obtained before his death.M C Makris, K C Koumarelas, and A S Mitrousias wrote the paper, M C Makris and E Yettimis designed, supervised, and edited the paper, G G Psathas and A Mantzioros performed literature search and data extraction, and S P Sakellariou and P Ntailiani performed immunohistological examination of the specimen.


The development of various benign oral mucosal lesions associated with dental implants, such as pyogenic granuloma or peripheral giant cell granuloma, has been rarely reported. However, the occurrence of vascular diseases, such as hemangioma, related to dental implants has not been explored in the literature. In this study, we report a case of co-development of pyogenic granuloma and capillary hemangioma on the alveolar ridge associated with a dental implant in a patient undergoing antithrombotic therapy. To the best of our knowledge, this is first case of hemangioma formation associated with a dental implant.A 68-year-old Korean man was referred for intermittent bleeding and a dome-shaped overgrowing mass on his upper alveolar ridge. He underwent dental implantation 5 years ago, and was started on warfarin for cerebral infarction a year ago. He had experienced gum bleeding and gingival mass formation 6 months after warfarinization; then, his implant fixture was removed. However, his gingival mass has been gradually increasing. The gingival mass was surgically excised, and revealed the coexistence of pyogenic granuloma and capillary hemangioma in histological analysis of the specimen. The lesion has showed no recurrence for more than a year.Regarding immunostaining features, the endothelial cell markers, CD34 and CD31, and the mesenchymal cell marker, vimentin, were strongly detected, but cell proliferation marker, Ki-67, was negatively expressed in the endothelial cells of the hemangioma portion. However, in the pyogenic granuloma portion, CD34 was almost negatively detected, whereas vimentin and Ki-67 were highly detected in the fibroblast-like tumor cells. According to these heterogeneous characteristics of the lesion, the patient was diagnosed with coexistence of pyogenic granuloma and capillary hemangioma associated with the dental implant on the attached gingiva. We recommend that patients with dental implants who have chronic peri-implantitis under antithrombotic therapy should be closely followed to ensure early detection of oral mucosal abnormalities. Dental implantation has become one of the most common dental procedures. However, some serious complications related to dental implant placement, including hemorrhage and life-threatening swelling, have occasionally been reported . In addiIn medically compromised patients, some unpredicted complications can be caused by simple dental procedures, such as tooth extraction or dental implant placement. In particular, antithrombotic therapy could induce postoperative bleeding or hematoma formation. However, the development of any vascular disease, including hemangioma or vascular malformation, in patients undergoing antithrombotic therapy has not been explored in the literature. Hemangioma is a benign tumor of vascular endothelial cells, characterized by the benign proliferation of blood vessels, and is the most common type of childhood tumor ,8. This In this study, we report a case of co-development of PG and capillary hemangioma in the upper alveolar ridge associated with a dental implant in a warfarinized patient. To confirm the characteristics of the lesion, the excised specimen was immunohistochemically analyzed with various antibodies for markers of endothelial cells, mesenchymal cells and cell proliferation. To the best of our knowledge, this is first case of hemangioma formation associated with a dental implant in a medically compromised patient.A 68-year-old Korean man was referred by his dentist for intermittent bleeding and a dome-shaped overgrowing mass on his upper left alveolar ridge Figure A. He undHistology of the specimen revealed an ulcerated nodule and edematous granulation tissues under the epithelium of the tumor with numerous small blood vessels and neutrophil infiltration. The patient was diagnosed with PG Figures A and 2B.The causes of reactive peri-implant mucosal diseases have not been completely understood. One putative cause is the corrosion of the fixture surface and metal particles . In addiHemangioma is one of the most common tumors in neonates and children, and is classified into capillary and cavernous types on the basis of the size of vascular spaces and histology. Capillary hemangioma is characterized by numerous proliferating small thin-walled blood-filled vessels composed of a single layer of endothelial cells, surrounded by a discontinuous layer of pericytes and reticular fibers, and by its uncommonness in older aged people ,8. HowevIn this report, the patient had used the prosthesis after implantation for 4.5 years, and undertaken antithrombotic therapy with warfarin for a year. He felt an abnormal gingival mass formation and spontaneous gum bleeding starting 6 months after warfarinization. Interestingly, the gingival mass showed two different entities, PG and capillary hemangioma, in terms of its histological features. In the immunohistochemistry of the hemangioma portion of the lesion, hematopoietic markers, CD31 and CD34, were highly detected in neogenerated vascular tissues, whereas the cell proliferating marker, Ki-67, was negatively detected in the endothelial cells of the hemangioma. However, almost negative expression of CD34 but strongly detected Ki-67 was observed in the PG portion. The mesenchymal cell marker, vimentin, was strongly detected in both endothelial cells of the hemangioma and fibroblasts in the PG portion. The results of the immunohistochemical analysis of the present case indicate that these histomorphologically different entities possess different properties and have developed coincidentally in one gingival mass.Although a direct relationship between hemangioma development and warfarinization could not be established in this case, frequent bleeding due to antithrombotic therapy and chronic inflammation of peri-implantitis are suspected to be causally associated with the development of neovascularization as well as PG in the gum tissue. Chronic irritation, including inflammation of peri-implantitis, could generate various neoplasms in the oral mucosa ,19. To tWritten 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.The authors declare that they have no competing interests.YHK collected and analyzed the patient data, and was a partial contributor in writing the manuscript. JHB and MJC assisted treatment of the patient and performed patient care. JSL evaluated the histological and immunohistochemical results of the tumor specimen. JHJ and YIK referred the patient to our clinic and offered the old data when the patient had undertaken dental implantation. BWP performed the patient management, and was a major contributor in writing the manuscript. All authors read and approved the final manuscript.


Laparoscopic cholecystectomy is one of the most frequently performed surgical interventions nowadays in developed countries. While lost gallstones during the procedure represent a commonly encountered issue, there is an ongoing debate whether split gallstones imperatively need to be extracted during the same procedure. The reported case of a wall abscess several years after follow-up lights up this debate.A 75-year-old male Caucasian with a history of rheumatoid arthritis and congestive heart failure presented with a recurrent subcutaneous abdominal wall abscess with occasional, spontaneous drainage of pus. He underwent laparoscopic cholecystectomy for acute calculous cholecystitis 3 years ago with uneventful and prompt recovery. A computed tomography scan showed a cavity in the periumbilical abdominal wall with peripheral contrast-enhancing, next to a calcified foreign body between the rectus muscle sheets. Wound exploration under general anaesthesia was performed with drainage of the cavity, extraction of the foreign body and closure of the anterior rectus sheet over a drainage catheter. The foreign body turned out to be a gallstone lost in the periumbilical port site during the procedure. Antibiotic treatment with co-amoxiclav was continued for 14 days. The patient was discharged 9 days postoperatively with a clean wound.This case and short review of the literature is a reminder of the importance of careful extraction of split gallstones during cholecystectomy in order to avoid early or late complications. This is especially important in the light of one of the most commonly performed surgical procedures in developed countries with generally low morbidity. Split gallstones during laparoscopic cholecystectomy are a well known issue when the gallbladder containing the calculus is accidentally perforated during the procedure. An Australian group reviewed eight studies with over 18,000 performed laparoscopic cholecystectomies and found an incidence of perforation of the gallbladder of 18.3% (8–39.9%) and an incidence of spilt gallstones of 7.3% 0.1–20%), while one third (5–48%) of the split gallstones were left unretrieved [0%, whileE. coli” bacteria were found on microbiological array. Antibiotic treatment with co-amoxiclav was continued for 14 days. The patient was discharged 9 days postoperatively with a clean wound, and continued to visit the outpatient clinic for wound follow-up for 8 weeks. Follow-up was uneventful.A 75-year-old male Caucasian with a history of rheumatoid arthritis and congestive heart failure presented with a recurrent subcutaneous abdominal wall abscess with occasional, spontaneous drainage of pus. He underwent laparoscopic cholecystectomy for acute calculous cholecystitis 3 years ago. Physical examination revealed periumbilical redness and tenderness with a draining percutaneous tract. Laboratory testing revealed a white cell count of 13,900 per cubic millimetre. A computed tomography (CT) scan showed a cavity in the periumbilical abdominal wall with peripheral contrast-enhancing, next to a calcified foreign body between the rectus muscle sheaths (Fig. Since over 90% of split gallstones never become symptomatic, they often present as incidental findings on CT-scans. Particular locations such as Morison’s pouch or even intrathoracic stones have been described , 5. “LooInformed patient consent with mentioning of this potential late complication might help to avoid misunderstandings later on. Of note, interventional abscess puncture without stone removal implies a high risk of recurrence .The recently encountered case of an abdominal wall abscess due to an infected gallstone, split during a surgical intervention several years ago, is a reminder of this displeasing late complication. It reminds the surgical community of the importance of prompt and careful removal of these stones, especially when confronted with acute cholecystitis. Treating physicians confronted with unusual abscess locations should bear in mind this rare complication when confronted with an unusual abscess formation in patients who have previously undergone laparoscopic gall bladder removal.Written informed consent was obtained from the patient for publication of this Case Report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.


With advances in surgical procedures and perioperative management, hepato-biliary-pancreatic surgery, including hepatectomy and pancreaticoduodenectomy, has been employed for recurrent colon cancer. However, no report has described a case of major hepatectomy with the combined resection of hepaticojejunostomy following pancreaticoduodenectomy for locoregionally recurrent colon cancer. Here, such a case is reported. The patient, a 37-year-old woman, had undergone pancreaticoduodenectomy for lymph node recurrence along the extrahepatic bile duct from cecal cancer. Thirteen months later, a biliary stricture was found at the hepaticojejunostomy site and right hepatectomy was performed. The resected specimen showed a papillary tumor at the hepaticojejunostomy. Based on its histological features, the pathogenesis of this tumor was considered to be intramural recurrence via lymphatic vessels. Although she underwent resection of a lymph node recurrence at her mesentery 12 months later, she has remained well thereafter, without any sign of further recurrence during 5 years of follow-up after hepatectomy. The management of locoregionally recurrent colon cancer poses a surgical challenge since these lesions often extend into surrounding structures and organs. Surgery is the sole curative modality that can result in long-term survival after complete resection alone . UsuallyThe patient was a 37-year-old woman who had undergone right hemicolectomy for cecal cancer at a different hospital. Histological examination revealed a well-differentiated adenocarcinoma with mucinous carcinoma, which had invaded the serosal layer without regional lymph node metastasis. A slight lymphatic and vascular invasion was evident. Finally, her tumor was diagnosed as T4aN0M0, stage II B according to UICC classification  Figure . Adjuvanen bloc resection of the right hepatic and caudate lobes and the anastomotic region of the bile duct and jejunum. The jejunum around the anastomosis was resected with a 1-cm margin and closed by suturing transversally  has been employed for advanced biliary tract cancer with relatively low morbidity and mortality ,7. MoreoIn the current case, the anastomotic tumor may have been the result of intramural recurrence via the lymphatic vessels because tumor invasion to the lymphatic vessels was observed in both the metastasized lymph node that was resected with PD and the anastomotic tumor . We considered two other possible explanations of the pathogenesis. The first relates to the development of the second primary cholangiocarcinoma after bilioenteric anastomosis. Repeated chronic inflammation due to regurgitation from anastomosis evokes epithelial damage, resulting in carcinogenesis, and is the only independent predictor of the development of cholangiocarcinoma . HoweverIn the present case, a renewed hepaticojejunostomy was created between the left hepatic duct and the inverted blind end of the jejunum. However, in all previously reported cases, bilioenteric continuity was reestablished using the lower jejunum. At the previous surgery, the lifted jejunum and its mesentery (for bilio- and pancreato-enteric reconstruction) were usually fixed to the mesentery of the transverse colon in order to prevent internal hernia. Therefore, the lower jejunum was unlikely to have enough length to complete the renewed hepaticojejunostomy safely. Our method appears to be advantageous when the blind end of the jejunum is sufficiently long.We have reported a case of a tumor at hepaticojejunostomy that was successfully resected with right hemihepatectomy 2 years after pancreaticoduodenectomy for lymph node recurrence of cecal cancer. The pathogenesis of the anastomotic tumor was considered to be intramural recurrence via lymphatic vessels. Aggressive surgery should be performed to achieve long-term survival in cases of locoregionally recurrent colon cancer, even if the surgery is technically demanding.Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.


The duodenum is infrequently affected by malignant lymphoma, and follicular lymphomas of the duodenum are rare histological subtypes. There are no reported cases of collision of follicular lymphoma and other tumors in the ampulla of Vater. A 57-year-old Japanese man presented with jaundice, and abdominal computed tomography revealed a tumor of the ampulla of Vater invading the pancreatic head with biliary dilatation and a thickened duodenal wall. The patient underwent subtotal stomach-preserving pancreaticoduodenectomy. Histopathology of the resected specimen revealed lymphoid follicular formations with small-to-medium-sized centrocyte-like cells and some centroblast-like cells. The atypical lymphoid cells were immunohistochemically positive for CD10, CD20, and CD79a but negative for CD5 and cyclin D1. BCL2 protein was highly expressed in the follicle centers. The diagnosis was duodenal follicular lymphoma, Grade 1. The follicular lymphoma, 40 mm in diameter, involved duodenal submucosa and regional lymph nodes without distant metastasis. This duodenal follicular lymphoma was partially overlapped by adenocarcinoma of the ampulla of Vater, measuring 25 × 20 mm, which involved the lower common bile duct, pancreas, and duodenum. We report the first case of a surgically treated collision tumor composed of a rare mass-forming follicular lymphoma and adenocarcinoma of the ampulla of Vater. Gastrointestinal (GI) primary lymphomas are the most frequent extranodal lymphomas, accounting for 30–40% . The stoWe report the first unique case of a collision tumor, in which a rare mass-forming FL was incidentally diagnosed from a surgically treated specimen of cancer of the ampulla of Vater.γ-glutamyl transpeptidase (γGT) 1472 IU/L , total bilirubin 15.3 mg/dL , and direct bilirubin 11.6 mg/dL . CA 19-9 was 101.7 U/mL , while CEA was within the normal range. Contrast-enhanced abdominal computed tomography (CT) revealed (i) dilatation of the biliary tree, (ii) tumor of the papilla of Vater invading the pancreatic head, measuring 18 mm in diameter, and (iii) a thickened duodenal wall. Neither lymph node enlargement nor splenomegaly was demonstrated. Contrast-enhanced magnetic resonance imaging (MRI) revealed an irregular tumor corresponding to the ampulla of Vater with infiltration to the adjacent duodenal wall, involvement of the lower bile duct, and dilated intrahepatic and extrahepatic bile ducts, including the cystic duct and the gallbladder. The common bile duct (CBD) measured 15 mm in diameter with an abrupt concentric stenosis in its lower third and a cut-off point located 20 mm distally to the duodenal ampulla  128 IU/L , alanine aminotransferase (ALT) 232 IU/L , alkaline phosphatase (ALP) 647 IU/L ,  ampulla . The panThe patient underwent endoscopic retrograde cholangiopancreatography (ERCP), and a nasobiliary drainage tube was placed. The duodenal papilla was depicted as being covered with whitish tiny granules, whereas the tumor of the ampulla of Vater was not exposed . Biopsy The tumor of the ampulla of Vater was macroscopically observed as an ulcerative mass accompanied by surrounding multiple small nodular lesions Figures  and 4.Histopathological study of the tumor unexpectedly revealed that the lesion was composed of two independent neoplasms. The surface part of the tumor showed lymphoid follicular formations  with smaThe patient was discharged one month after the operation without critical complications. S-1 oral intake was selected as adjuvant chemotherapy targeting the cancer of the ampulla of Vater. Nine months after the operation, multiple liver metastases were detected. CT scans suggested that those lesions were derived from adenocarcinoma of the ampulla of Vater rather than duodenal FL. The patient received systemic chemotherapy including gemcitabine; however, multiple lung metastases with pleural effusion and peritoneal dissemination appeared. Metastatic adenocarcinoma was cytologically suggested in the pleural effusion, whereas involvement of FL was not suggested. One year after the operation, the patient died of multiple organ failure with primary disease progression. Autopsy was not permitted.Previous studies characterized primary duodenal FL as a markedly indolent FL variant, which, even when left untreated, very rarely disseminates and does not transform to high-grade disease , 7, 12. Diagnosing FL of the duodenum is not easy , 5. In aCareful clinical examination, including the capsule of double-balloon enteroscopy, seems to increase the number of cases diagnosed as duodenal FLs –7. EndosThe presenting symptoms leading to gastroduodenoscopy were not related to the duodenal FL of reported cases , 6, 10. There is no reported case of a collision tumor in the ampulla of Vater, composed of FL and other tumors, and its tumorigenesis is uncertain. Niu et al.  hypothesConcerning carcinogenesis, a high incidence of biliary tract cancer has been revealed, particularly in pancreaticobiliary maljunction . A mixtuCollectively, these hypotheses could support the cause of the collision tumor in our case. For example, direct stimulation of bile and pancreatic juice in the duodenal mucosa might affect both lymphocytes and the epithelium of the ampulla of Vater. Development of FL, for example, forming a relatively large mass lesion as a rare form as in our case, might decrease the local immunodefense system and facilitate the tumorigenesis of adenocarcinoma from the affected epithelium of the ampulla of Vater. In this line, the tumorigenesis of only one reported case  of a synWe herein present the first unique case of symptoms due to cancer of the ampulla of Vater, whereas FL, an asymptomatic and infrequent tumor, was incidentally diagnosed as part of a collision tumor in a resected specimen of the cancer.


Three cases of intracranial angioleiomyoma (ALM) in our neurosurgery center are reported in detail. ALM is a benign soft tissue tumor comprised of mature smooth muscle cells and a prominent vascular component, which is extremely rare as a primary intracranial lesion. Altogether, only 12 cases were recorded in the literature to date, to the best of our knowledge. Case 1 is the second report of intra-sella ALM, a 51-year-old woman presented with visual deterioration for 2 months. An MRI showed an intra-sella 3-cm tumor, partially flame-like, enhanced with gadolinium. Using microscopic endonasal transsphenoidal approach, the tumor was completely resected with great difficulty. At 11 days post-surgery, she died of a sudden catastrophic nasal hemorrhage. An angiography revealed a pseudo-aneurysm of ICA . Case 2 is a 49-year-old man who presented with weakness of the lower limbs for 1 year. A large subtentorial mass was found affixed to the torcular and straight sinus, which was partially flame-like, dramatically enhanced as in case 1. Case 3 is that of a 77-year-old man. An ALM mass was revealed in the diploe of left temporal bone, and had eroded the inner table. Immunohistochemical workup confirmed the diagnosis of angioleiomyoma in all 3 cases. The radiology, operation, and complication of surgery in each case were discussed. In conclusion, intracranial ALMs are extremely rare, usually located ex-neuro axis (such as in our cases), in the sella, in posterior fossa, or in the skull. Magnetic resonance imaging (MRI) revealed a special feature of flame-like partial enhancement that may be helpful to distinguish ALM from pituitary tumors or meningiomas, and should result in the consideration of this rare tumor entity early on in the diagnostic process. A definitive diagnosis depends on histological analyses. The resection of ALM in certain locations is difficult and risky because of the rich blood supply. Angioleiomyomas (ALMs) are benign soft tissue tumors commonly occurring in middle-aged women as nodules in the subcutaneous tissue of the lower extremities, which iOur center admitted about 4403 cases of primary intracranial tumor in the last 5 years, only 3 of which (less than 0.1%) were identified as ALM. Case 1 is the second report of intra-sella ALM (the first was reported in 2010), case 2 A 51-year-old woman presented to our neurosurgery center with a two-month long blurry of the right eye, which gradually worsened, eventually losing vision in the eye 20 days before admission. A neurological examination revealed that the sight of the right eye was 0.03, with bilateral temporal hemianopsia. There were no other remarkable signs. An endocrinological evaluation showed only a low value of blood cortisol. T1-weighted (T1-w) MRI scans revealed an iso-signal intra-sella tumor sized 3.0 × 2.5 × 2.5 cm, close to the medial part of the right cavernous sinus, with distinctive partial enhancement after a gadolinium injection, which arose from the right cavernous sinus, flame-like in shape, with bush-like edge , arteriovenous malformation (AVM), cavernous hemangioma, angiofibroma (AFM), and myopericytoma (MPC). As an infiltrating high-grade glioma, GBM is often a necrotic and cystic lesion, with high metabolism and high proliferation ability. It is usually GFAP-positive, has no diffused vessel structure, and mitotic figures can be seen. In our ALM cases, after meticulous searching, no mitoses was found, the Ki67-/MIB-index was very low, and GFAP was always negative. AVMs are described as abnormalities consisting of tangled masses of tortuous arteries, veins, and abnormal connecting channels. In the cavernous hemangioma we can also see extremely distant blood vessels or a blood-filled sinus. In ALMs, CD34 is positive in the vessel endothelium, SMA is positive in the smooth muscle bundles, and (80 to 90% of cases) desmin is also frequently immuno-positive. Angiofiet al.[Although ALM is benign and seemed to be easily curative by surgery in the literature, we have to say, for the abundance of vascular structure, surgery on ALM in certain locations is difficult and risky, especially when the tumor is rather large. In case 1, the operation was difficult and the delayed fatal complication was extremely rare. The reason of the formation of the pseudo-aneurysm was not clear, lessons should be drawn from the catastrophic outcome. The tumor might have eroded the wall of ICA, the risk of potential postoperative hemorrhage should be considered, and an earlier angiography could have avoided the fatal outcome. In case 2, as a large vascular mass in the posterior fossa, there was heavy blood loss in order to excise it piecewise. Figueiredo et al. also repIntracranial ALMs are extremely rare and are usually located on the ex-neuro axis, such as in our cases . MRI scans revealed a special feature of flame-like partial enhancement that may be helpful to distinguish ALM from pituitary tumors or meningiomas, and should result in the consideration of this tumor entity early in the diagnostic process. A definitive diagnosis depends on a histological analyses. The resection of ALM in certain locations is difficult and risky because of the rich blood supply.Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the editor-in-chief of this journal.ALM: Angioleiomyoma; AVM: arteriovenous malformation; CT: Computed tomography; DSA: Digital subtractive angiography; FLAIR: fluid-attenuated inversion recovery; GBM: glioblastoma multiform; GFAP: glial fibrillary acidic protein; ICA: internal caratid artery; NF: neurofilament; H&E: Hematoxylin and eosin; JNA: juvenile nasopharyngeal angiofibroma; MPC: Primary intracranial myopericytoma; MRI: Magnetic resonance imaging; PVA: polyvinyl alcohol; SMA: Smooth muscle actin; Vim: vimentin.The authors declare that they have no competing interests.LS reviewed the literature and drafted the manuscript. YZ and LS were clinically responsible for patient care. HW directed the operation and revised the manuscript. All authors read and approved the final manuscript.


Solitary fibrous tumors of the pelvis are rare. We report the case of a 32-years-old patient who presented with abdominopelvic mass. The imaging studies showed a right adnexal mass of more than 10 cm. Exploratory laparotomy revealed a 20 cm mass at the Douglas pouch which was adhered to the posterior wall of the uterus. Complete resection of the mass was performed. Histological analysis showed a spindle cell undifferentiated tumor whose morphological and immunohistochemical profile are consistent with solitary fibrous tumor. It is important to know that although these tumors are rare, their evolution can be pejorative. Therefore, long-term followup should be recommended. Solitary fibrous tumors (SFTs) are rare mesenchymal tumors which can be benign or malignant. They occur most often in the pleura, but have also been described in many extrapleural locations. Pelvic localization is very rare.We first report a case of solitary fibrous tumor revealed by abdominopelvic mass in a 32-year-old woman, and then we describe the clinical, radiological, morphological, and immunohistochemical characteristics of this condition.Mrs. BY, 32-year-old woman, with no medical and family history, gravida 2 para 2, with regular menstrual cycles, using intrauterine contraceptive device (IUD) for three years, was referred by her general practitioner for abdominopelvic mass associated with pelvic pain lasting for one month.Physical examination showed a 20 cm abdominopelvic mass lateralized to the right, which is regular and very mobile. No lymph nodes were palpated. The pelvic exam found a right adnexal mass that appears to be independent of the uterus. The rest of the physical examination was normal.Pelvic ultrasound with abdominal and endovaginal transducers depicted a right adnexal mass measuring 20 cm, which is heterogeneous, with tissular and cystic components and without intracystic or extracystic exophytic vegetations and without peritoneal fluid. The peritoneal space was free of other abnormalities . The detPelvic MRI showed the presence of an adnexal mass, measuring 25/20 cm, which seemed to grow at the expense of the right ovary. The signal was isointense on T1-weighted imaging (WI), with a heterogeneous appearance with alternating hyper- and hypointense zones on T2-WI, as well as a heterogeneous enhancement after gadolinium injection. This mass had a mixed solid and cystic composition with enhancement of the solid parts. The cystic component contains multiple cysts with septations measuring from few millimeters to 7.5 cm. The left ovary appeared normal and there are no deep lymph nodes or pelvic effusion . LaparotOnce the diagnosis was established, a chest CT was performed in order to rule out a primary tumor and no abnormality was found.The patient was followed by clinical and imaging examinations for the following four months.The term solitary fibrous tumor (SFT) is the current name to designate a tumor proliferation of spindle cells of mesenchymal origin. It was initially described in the pleura in 1931 by Klemperer and Rabin . This tuThe pelvis is a rare localization of SFT . It occuThe ultrasound examination is carried out in a complementary first line; it allows for the specification of the characteristics of the mass with Doppler vascularity. Pelvic ultrasound of our patient depicted a right adnexal mass measuring 20 cm, which is heterogeneous, with tissular and cystic components and without intracystic or extracystic exophytic vegetations and without peritoneal fluid.MRI usually shows an isosignal on T1-WI with a heterogeneous signal combining hyper-and hypointense zones in a patchy pattern on T2-WI, and a heterogeneous enhancement after gadolinium.Macroscopically, they are whitish large masses of 10 cm (long axis), well delineated but not always encapsulated. In our observation, macroscopic examination found a white, smooth, and bumpy mass, measuring 25/22 cm, which is concordant with the literature.Diagnosis of SFT can be difficult because of their unusual location and variability of pathological profile. Histological examination shows alternating areas of hypocellularity and hypercellularity along with thick bands of hyaline fibrosis. Cells can be round or fusiform. On immunohistochemistry, the majority of SFTs are positive for CD34 , 8, 9 anThe evolution of these SFTs is often slow and painless, except in the case of large tumors that may cause various cuts. Between 10 and 15% of SFTs are aggressive malignant seen with metastasis of the lung, liver, and bone. SFTs are often malignant. Hypercellular atypia shows moderate to marked cytonuclear with foci of tumor necrosis and numerous mitoses (more than four in ten fields at high magnification) , 10. OurExtrapleural SFTs are slowly progressing tumors with a low recurrence rate of 2 to 15%. The latter is related to the quality of the surgical resection and rare malignancy of extrapleural location , 6, 8. AA recent in vitro study showed that a solitary fibrous tumor with a mitotic index of 7/50 HPF benefits from chemotherapy, including 5-FU, adriamycin, mitomycin-C, and docetaxel. These treatments could benefit solitary fibrous tumors which are more aggressive, recurrent, unresectable, or with incomplete resection .Pelvic solitary fibrous tumors are rare and most often benign as described in other localisations. However, malignant transformation is possible and can be correlated with some histological criteria. No consensus currently exists regarding these SFTs, but care must be maximized. No study has shown the effectiveness of additional treatment with chemotherapy or radiotherapy. Finally, a long-term followup is recommended for those patient who are at risk of relapsing.


Dermatofibrosarcoma protuberans is an uncommon low-grade soft tissue sarcoma with a high potential for recurrence as it has irregular finger like extensions.We report a case of a large, recurrent dermatofibrosarcoma protuberans in a child involving the anterior abdominal wall, which posed a challenge for reconstruction. Peritoneum sparing full thickness resection of the anterior abdominal wall, meshplasty and a free anterolateral thigh flap was performed for reconstruction of the defect.Large composite defect, involving more than half of the anterior abdominal wall, necessitate a free flap reconstruction. Although these reconstructions are technically challenging in children, they are the only option available.Complete surgical excision is essential for DFSP of the abdominal wall, which may result in large challenging defects. Free flaps remain the only option in this scenario and hence it is essential to have expertise for microvascular flap reconstruction. Dermatofibrosarcoma protuberans (DFSP) is an uncommon, low grade soft tissue sarcoma of fibroblast origin. Surgical excision with negative resection margins is crucial to prevent recurrences . The skin with the linear scar, hatch marks and the drain site scar were included in the resection Figure . Since tm Figure . A free m Figure . The deem Figure .Figure 3The patient had an uneventful recovery and was discharged from the hospital on the eighth postoperative day. All surgical resection margins were negative. The closest margin was 2 mm microscopically although grossly it was 12 mm away. The patient received postoperative radiotherapy . At first follow up after three months the flap is well healed and the patient is disease free.DFSP is a locally aggressive tumor with a high potential for recurrence as it has irregular finger like extensions (Bichakjian et al. Kawaguchi et al.  had suggComplete surgical excision is essential for DFSP of the abdominal wall which may result in large challenging defects. Free flaps remain the only option in this scenario and hence it is essential to have expertise for microvascular flap reconstruction.


Myeloid sarcoma results from the extramedullary homing and proliferation of immature myeloid precursors. We present the timeline, events and diagnostic pitfalls related to a 66 year-old male patient’s case, admitted to the Hematology Clinic for pancytopenia, fever, weight loss and fatigue. The severe cytopenia and the few blasts observed in his blood smear indicated a bone marrow biopsy. The bone marrow showed hypercellularity and multilineage dysplasia with the presence of 15% myeloblasts. After the biopsy, he promptly developed paraplegia and nuclear magnetic resonance revealed an epidural tumour which was then resected.In the epidural tumour mass blast-like, round cells were observed with a complex immunophenotype, characterized by myeloperoxidase, CD117, CD15, CD99, leucocyte common antigen positivity and a high Ki-67 proliferation index. Considering the main differential diagnostic issues, the final diagnosis was stated as myelodysplastic syndrome-associated myeloid sarcoma. The prognosis was unfavourable, the bone marrow was quickly invaded by proliferating blast cells, and despite chemotherapy attempts, the patient died. Every anatomical site of the body can be involved, but most common are the soft tissues of the head and neck, bone, skin and less often the genitourinary tract, the central nervous system and the spinal cord3. We present a case with a complex clinical manifestation resulting from the unusual localization of the tumour mass, and that coincides with the MDS involving simultaneously the bone marrow tissue.Myeloid sarcoma is a rare malignant solid tumour resulting from the extramedullary proliferation of myeloblasts or immature myeloid cells, that usually precedes or accompanies acute myeloid leukaemia (AML), chronic myeloproliferative disorders, sometimes presenting as the first sign of AMLAn otherwise healthy, 66-year-old man was admitted to the Haematology Clinic of the Emergency County Hospital  in a poor general condition, presenting fatigue, continuous fever, night sweating and weight loss (10 kg in 3 months), with symptoms occurring for a few weeks. His past medical history was unremarkable, without a known history of toxic or drug exposure. Physical examination evidenced a mucocutaneous pallor, but no lymphadenopathy or organomegaly. Acute myeloid leukaemia/myelodysplastic syndrome was suspected. Baseline laboratory data confirmed symptoms of ineffective haematopoiesis. Peripheral blood examination revealed pancytopenia: severe anaemia, trombocytopenia and leukopenia with 2% blasts of myeloid lineage , associaThe hypercellular bone marrow biopsy showed a multilineage dysplasia characterized by dyserythropoiesis, dysgranulopoiesis and dysmegakaryopoiesis with approximately 15% CD34/CD117 positive myeloblasts without Auer rods, associated with the presence of abnormal localisation of immature precursors (ALIP). Based on this histologic picture, according to the clinical and laboratory data, our diagnosis was refractory anaemia with excess blasts-2 (RAEB-2). The spinal tumour fragments showed a diffuse proliferation of undifferentiated myeloblast-like round tumour cells, mixed with a few neutrophil precursors.Considering the two histological findings, myeloid sarcoma was suspected, but for a differential diagnosis lymphoblastic lymphoma, Burkitt lymphoma, diffuse large B-cell lymphoma and small round cell tumours also had to be excluded. A large immunohistochemistry panel was helpful in achieving differentiation of these conditions. The monomorphic tumour cells revealed positive stainings for Leucocyte Common Antigen (LCA), myeloperoxidase (MPO), CD15 and CD117, associated with a high Ki67 index (over 60%). Neuroendocrine and lymphoid markers, pan CK and also CD68 were negative. In contrast, a significant number of tumour cells expressed CD99 antigen.Myelodysplastic syndrome (RAEB-2) associated myeloid sarcoma” of the spinal cord.The histopathological diagnosis has been stated on the basis of the two biopsies, being completed to “The clinical course was unfavourable, at the end of the first week post-surgery, the peripheral blood and bone marrow suddenly showing a burst of the blast count up to 50%. Chemotherapy with high-dose cytosine arabinoside proved to be inefficient, and the patient died at 5 weeks from admission.1. The first description of “myeloid sarcoma” appeared in literature in 1811, and in 1904 its association with acute leukaemia was identified4.The exact prognosis of myeloid sarcoma is difficult to determine, but in general is poor5.The term "granulocytic sarcoma” was introduced in 1967 by Rappaport to describe only tumours of granulocytic origin, however the term is now often applied to any tumour related to acute leukaemia or myelodysplastic syndrome (MDS)5.The mechanisms of extramedullary involvement are not fully understood, but it was demonstrated that leukaemic cell surface markers (CD56) are implicated in extramedullary hominget al. analyzed 12 cases of myeloid sarcoma and found a consistent positivity for CD117, CD43, MPO, CD68, CD34 and a sporadic reaction of leucocyte common antigen6. Two major issues of the correct diagnostic orientation are the recognition of a possible non-lymphoid CD45 positivity and ruling-out of the aberrant CD99 antigen from the pivotal markers in this tumour type. On the other hand, MPO positivity is a prominent feature for a tumour of myeloid origin that overrides the unusual and aberrant antigen expression .The particularity of our case consists of the unusual tumour localization with spinal compression and neurologic signs, and the combination with the deficient haematopoiesis, that raises the suspicion of a metastasis in the bone marrow. Regarding phenotype, the positivity for CD45 and CD99 with a high Ki-67 proliferation index could suggest a lymphoma, but the morphological similarities between the extramedullary tumour cells and the bone marrow blasts are suggestive for myeloid sarcoma. For an accurate approach, a large-scale immunohistochemistry panel, containing mandatorily the CD45, MPO, CD15, CD79a, CD3, CD34, CD117, CD99, TdT and Ki-67 proved to be useful. The major diagnostic pitfalls are represented by distinction from lymphoblastic lymphomas (TdT+/CD34+/CD99+/CD45+/high Ki-67 index), and small round-cell tumours (CD99+/TdT-). SeifertIn conclusion, we considered, that a large panel of antibodies must be used to establish the challenging diagnosis of myeloid sarcoma. This condition that should always be considered as a diagnostic possibility in patients with MDS, AML or chronic myeloproliferative disorders complicated with a secondary tumor mass.Written informed consent was obtained from the patient for the management and publication of clinical and laboratory data. The Ethical Committee of the Hospital approved the publication of the clinical details (no. 780/14-01-2016). Well-structured paper. Rare case, nice demonstration of the difficult differential diagnostic process of myeloid sarcoma. The title is appropriate and represent the work. Results supports the final conclusion of the case.I have read this submission. I believe that I have an appropriate level of expertise to confirm that it is of an acceptable scientific standard. The main aim of this short case report is to create attention for myeloid sarcoma, which can be a difficult differential diagnostic problem, first of all in cases, when the myeloid sarcoma precedes the bone marrow disease. The documentation of the presented patient illustrates well the histological work up and the diagnosis, but the paper would have been more teaching if the authors add more detail about the patients course. I would have been interested in the results of investigation of the cerebrospinal fluid, the cytogenetic findings and the neurological status after operation? Why monotherapy was chosen? What further complications did they observe? What was the cause of death?In my experience myeloid sarcoma can be purely blastic, but frequently shows signs of myeloid  differentiation. Fine cytoplasmic granulation of the immature cells, presence of eosinophilic myelocytes among the blasts, or some monocytoid features are suspicious for myeloid tumor, and can be recognised by careful examination of high quality sections and special stains. If these signs are observed a short panel containing the lineage specific markers is enough to confirm the diagnosis  For differential diagnosis of a poorly differentiated tumor, the authors list a wide panel of immune stains emphasizing markers of immaturity . I think the lineage specific markers, especially CD45, MPO, CD3, CD79, Pax5 and lysozyme, are more important. To suggest a cost effective algorithm in the report would be a valuable supplement.And I have to add, that the diagnosis of MDS with excess of blasts and AML is first of all based on bone marrow smears and flow cytometry. If flow cytometry is not available cytochemistry is recommended for defining the cell lineage of the blasts. Bone biopsy is usually only an additional investigation (except of dry tap). Probably in the presented case smears also were elaborated, but data of smears and bone marrow biopsy is not clearly separated. The blast count is based on CD34 and CD117 immunohistochemistry without mentioning the ratio of the blast by marrow smear cytology.  The authors state, that CD56 may have role in homing of the myeloid blasts, therefore additional CD56 investigation would be interesting. AML with t is more frequently cause myeloid sarcoma, therefore applying FISH probes to test the genotype of the tumor is also recommended to get more information about these rare tumors.I have read this submission. I believe that I have an appropriate level of expertise to confirm that it is of an acceptable scientific standard.


Myxoinflammatory fibroblastic sarcoma (MIFS) is a rare, painless, and intermediate (rarely metastasizing) fibroblastic tumor, which commonly occurs in the extremities, with an equal sex predilection. This sarcoma is composed of a mixed inflammatory infiltrate along with spindled, epithelioid, and bizarre tumor cells in a background of hyaline and myxoid areas. In spite of such a distinctive morphology, the tumor can be a diagnostic challenge, simulating inflammatory conditions as well as neoplastic nature. For accurate diagnosis, the tumor requires extensive clinical, radiological, and pathological investigations. We present a case of MIFS in a 19-year-old female who presented with a mass in the left ankle. After appropriate excision and postoperative radiation therapy, she is free of disease, including recurrence and metastasis, at 12 years postoperatively. Myxoinflammatory fibroblastic sarcoma (MIFS) is a rare intermediate (rarely metastasizing) fibroblastic tumor affecting males and females equally in the 40th and 50th decades of life. Described for the first time in three independent reports –3 in 199We recently have encountered a case of MIFS in the left ankle of a 19-year-old female. Her swollen left ankle was initially diagnosed as “tenosynovitis” by an open biopsy, although a few of atypical and multivacuolated cells were scattered in the lesion. Based on histopathology of an additional intralesional excision suggesting a low-grade sarcoma, she received reexcision and thereafter radiation therapy. Twelve years after the surgery, local recurrence was not found. Our case will provide useful information regarding accurate diagnosis and management of MIFS to orthopedic surgeons, pathologists, and radiologists, because over 10-year follow-up cases have not been reported.A 19-year-old Japanese female presented with painless swelling with local heat in the lateral aspect of her left ankle. She first noticed the swelling 3 months before this medical examination. She had no history of trauma on her feet and ankles. There was no skin color change overlying the mass. Her ankle Range of Motion (ROM) was unrestricted and full.μL) were within normal limits. Computed tomography (CT) scans and magnetic resonance imaging (MRI) showed an ill-defined heterogeneous soft tissue mass  in the subcutaneous tissue from the anterior to the lateral of the left ankle. There was a nodular or lobular mass showing T1WI low signal (Plain radiographs of the left ankle and foot showed unremarkable findings except for osteolytic lesion (about 5 mm in diameter) of the navicular bone. The results of routine laboratory tests including C-reactive protein level (0.2 mg/dL) and white blood cell count , epithelial membrane antigen (EMA), and neuron-specific enolase (NES). Percentages of p53- . At 41.4 Grays, skin flare was observed and radiation therapy was stopped. The patient was followed up by MRI  and boneAt the time of this writing (May 2015), the patient continues to remain disease-free after twelve years of surgical excision with no evidence of recurrence or metastasis.A new entity and an intermediate (rarely metastasizing) fibroblastic proliferation , 9, MIFSα-SMA, CD34, and S-100 protein. PCR-based assays for cytomegalovirus and Epstein-Barr-virus (EBV) are negative [Grossly, the tumor typically measures 1 to 5 cm, with the median size around 3 cm and mucoid character, and is usually poorly circumscribed , 7. Macrnegative , 7.The pathogenesis of MIFS is unknown. As noted in our case, no infectious agents, including cytomegalovirus (CMV) and EBV, have been identified , 6, 14. Natural history of the tumor is variable, with a high rate of local, often multiple, recurrences , 7. LocaIn conclusion, MIFS is a rare intermediate (rarely metastasizing) fibroblastic tumor and may easily be confused with many benign lesions. Although the clinical setting of MIFS is now distinctive, the initial diagnosis can be difficult to make. Therefore, surgeons, radiologists, and pathologists must include MIFS in their differential diagnosis of slow-growing tumors to avoid misdiagnosis leading to inappropriate management and delay in appropriate treatment.


Penile masses are a concerning finding for both patient and clinician upon initial presentation. There is a wide differential for penile masses from the benign  to more concerning malignant lesions. A proper history and physical is the first step to determining the etiology of the mass and any future clinical interventions. In this paper, we review a case of a 73-year-old male who is found to have an enlarging mass during work-up for possible placement of inflatable penile prosthesis. Fortunately, the mass was determined to be a benign epidermoid cyst presenting thirty years after reconstruction for Peyronie’s disease using dermal penile skin graft. With this unique presentation we review the scant literature on penile mass formation following Peyronie’s repair. In this particular case, we discuss Peyronie’s disease treated with plaque excision and dermal skin grafting.Peyronie’s disease is a common urologic entity with multiple options for definitive surgical repair. Plaque excision with grafting is a known and accepted method for reconstruction. There are multiple options for graft material, each carrying its own specific risk for complications and comorbiditiesA 73-year-old man was referred to our clinic in surgical consultation for possible placement of inflatable penile prosthesis due to progressively worsening erectile dysfunction. At his initial visit, he was found to have a non-tender rapidly growing mass in the distal penile shaft, which prohibited him from using his vacuum erection device. His past medical history was significant for type 2 diabetes and Peyronie’s disease. Thirty-two years prior, he underwent corrective surgery for Peyronie’s disease. Operative and clinical notes from that period could not be obtained, though the patient reported the procedure included plaque excision and use of a dermal penile skin graft. Following the procedure, he only reported mild residual penile desensitization.On exam, we noted a well-healed surgical scar and a 3 cm nodule arising from the left lateral aspect of the distal shaft. Moderate corporal fibrosis was also noted. We did not appreciate any concerning erythema, tenderness, drainage or ulceration. Importantly, the size and location of the mass prevented the patient from using a vacuum erection device. With the atypical presentation of this mass, the decision was made to proceed with further work-up prior to discussing any interventions for his erectile dysfunction.From this point, we proceeded with pelvic magnetic resonance imaging (MRI) without contrast . This rePenile duplex ultrasound, performed after an injection of 10 mcg alprostadil into the right corpora, revealed a 4 cm mass with complex internal echoes without Doppler flow. Compression of the corpora was seen with a moderate wasting deformity opposite of the mass. Approximately 15 degrees of mild leftward deviation was noted. Arterial peak flow was estimated at 12 cm/sec and resistive indices were 0.6 bilaterally. In addition, plaque without calcification was seen in the mid-shaft.Given the constellation of residual penile curvature, erectile dysfunction which was non-responsive to phosphodiesterase inhibitors, and the presence of a penile mass, the patient elected for placement of a three-piece inflatable penile prosthesis in conjunction with excision of the mass. We reviewed the possibility of other adjunct procedures such as penile modeling, grafting and plication. Penile prosthesis placement was declined by the patient’s insurance and mass excision was pursued alone. An incision was made over the site of the mass which was removed in its entirety without complication. During dissection, previous sutures from the dermal graft were appreciated. The corporal body was left intact. The pathology report was consistent with a benign inclusion cyst and his post-operative recovery was unremarkable with discharge to home immediately following surgery. Upon close follow-up, his penile curvature is stable, as determined by clinical exam and he has resumed using a combination of phosphodiesterase inhibitors and a vacuum erection device.There are multiple options for surgical management of Peyronie’s disease. Part of the treatment algorithm includes a number of options for grafting. It is well-known that skin grafts carry a greater risk of transplanting apocrine glands and hair follicles to the donor site. Due to this, it is incumbent upon the surgeon to pick a graft best suited for the operative site and graft intent. The surgeon must also weigh the risks and benefits of each possible donor site. Although this principle is followed in reconstructive surgery for Peyronie’s disease, there is a paucity of case reports documenting cyst formation after dermal graft inlay procedures. The authors most commonly use small intestinal submucosa or tunica vaginalis grafts.3. One case report describes a middle-aged male who presented with a unilateral enlarging penile lesion 2 years after having a dermal graft procedure for Peyronie’s disease. Upon exploration, a fluid filled keratin mass containing hair was removed2. The other case report described an elderly man who had dermal graft repair for a dorsal plaque3. The graft was harvested from a site devoid of hair, the abdominal wall near the left flank. Four years later the patient developed a swelling at the dorsum of the penis. In these two cases, the cyst presented less than 5 years after the operation.To our knowledge, there are two case reports describing this complication5. However, Peyronie’s disease is not thought to be a predisposition to a penile neoplastic lesion and to date the literature is devoid of a penile neoplasm after a dermal graft procedure6.In our case, the inclusion cyst presented more than 30 years after the operation, suggesting that cyst formation can be sporadic and yet rapid. The latency of cyst formation could be due to more extensive de-epithelialization of the graft in our case compared to the other cases. Still rapidly expanding soft tissue penile masses could be concerning for a neoplasm, albeit extremely rare. Therefore rapidly expanding soft tissue penile masses should be investigated with MRI to rule out a neoplasm and to further classify the lesion and location, which could prove to be valuable in surgical planningThe development of any penile mass should be concerning and warrants a full work-up by the appropriate medical provider. As this case shows, benign epidermoid cysts must be considered in those patients with a history of prior skin graft to the penis. Options for management of erectile dysfunction should not be limited following excision of an epidermoid cyst.Written, informed consent for publication of clinical details and images was sought and obtained from the patient. The authors provide a much needed perspective on penile masses in the setting of Peyronie's disease after surgical repair. The manuscript is well written and appropriately identifies an overall paucity of studies examining this complication, summarizing the extent of our knowledge in these specific situations in a total of 2 other available case reports. As such, this work is a very useful and important contribution to the literature. There are two points on which additional information would be useful, however. First, with regards to the patient case, it would be useful to include any serum studies that were performed, which may be salient particularly in the setting of a mass that is not benign. Second, a short discussion of the rarity  of malignant penile masses in this type of setting would be helpful.I have read this submission. I believe that I have an appropriate level of expertise to confirm that it is of an acceptable scientific standard. This is a well done article on a rare entity that urologists might encounter once or twice in a career.  Penile masses are certainly challenging and there is little data on their management.I have read this submission. I believe that I have an appropriate level of expertise to confirm that it is of an acceptable scientific standard.


In both cases, CT images revealed temporal bone defects and sclerotic changes around the tumor. Most parts of the tumor showed low signal intensity on T2-weighted MRI images and non-uniform enhancement on gadolinium contrast-enhanced T1-weighted images. No increase in signal intensity was noted in diffusion-weighted images. Dual-time-point PET/CT showed markedly elevated 18F-FDG uptake, which increased from the early to delayed phase. Nevertheless, immunohistochemical analysis of the resected tumor tissue revealed weak expression of glucose transporter-1 and hexokinase II in both tumors. Temporal bone tumors, showing markedly elevated 18F-FDG uptake, which increases from the early to delayed phase on PET/CT images, may be diagnosed as malignant bone tumors. Therefore, the differential diagnosis should include chondroblastoma in combination with its characteristic findings on CT and MRI.Temporal bone chondroblastoma is an extremely rare benign bone tumor. We encountered two cases showing similar imaging findings on computed tomography (CT), magnetic resonance imaging (MRI), and dual-time-point  Herein, we present two cases of temporal bone chondroblastoma, showing markedly elevated 18F-FDG uptake which increased from the early to delayed phase on PET/CT images, suggesting the need for caution against the misdiagnosis of these tumors as malignant tumors.However, to the best of our knowledge, there have been no reports on the features of this tumor, based on dual-time-point A 32-year-old male patient presented with hearing loss in the left ear. MRI was performed at a hospital to explore the cause of patient’s symptoms. As the images indicated, a left temporal bone tumor was suspected, and the patient was referred to our hospital for further evaluation and treatment.18F-FDG PET/CT imaging at our hospital  in the early (1h) and delayed phases (2h) were 17.1 and 20.2, respectively.Gadolinium contrast-enhanced T1-weighted images revealed non-uniform enhancement . No incr Figures  and 1i. The retention index (RI) was estimated at 18.1%, using the following formula:max-early SUVmax)/early SUVmax×100 (%)RI=(delayed SUVThe microscopic examination of biopsy specimens revealed that the tumor was mainly composed of uniform, round to polygonal cells and numerous randomly-distributed osteoclast-type giant cells. Considering the tumor site, the preoperative diagnosis was giant cell reparative granuloma, and complete tumor resection was performed. Histopathologic examination of the resected specimen revealed that the tumor contained a chondroid matrix and hemosiderin deposition, which suggested tumor hemorrhage .Few inflammatory cells, including lymphocytes, were infiltrated in the tumor tissue. Immunohistochemical analysis revealed S-100 positive cells . Conside18F-FDG PET/CT at our hospital (A 22-year-old male patient had undergone myringotomy several times within the previous year for recurrent otitis media with effusion in the left ear. MRI was performed at another hospital to explore the cause of recurrent episodes, and left temporal bone tumor was discovered. The patient underwent CT, MRI, and dual-time-point hospital .max values in the early and delayed phases on 18F-FDG PET/CT images were 17.3 and 19.4, respectively, and the RI was 12.1%. Tumor biopsy was performed. The preoperative diagnosis was a giant cell tumor, based on which the tumor was completely removed. The histopathological findings were similar to those in case 1 and the final diagnosis was temporal bone chondroblastoma.The findings of these imaging modalities were quite similar to those reported in case 1. However, in this case, signal intensity within the tumor in T2-weighted images was slightly lower than that reported in case 1. The SUVChondroblastoma is a rare, benign, cartilage-producing neoplasm, which is typically composed of round or polygonal chondroblasts, osteoclast-type giant cells, and a bluish to eosinophilic chondroid intracellular matrix with focal calcification . This tu18F-FDG uptake in spite of its benign pathological features (18F-FDG uptake (It is known that chondroblastoma often shows increased features . Hamada G uptake . Interes18F-FDG uptake in chondroblastoma is still unknown. However, considering the low signal intensity in T2-weighted MRI images and hemosiderin deposition in the resected specimen, tumor hemorrhage may contribute to some 18F-FDG pooling.Few lymphocytes were scattered; thus, inflammation is unlikely to be the cause of FDG accumulation. The precise mechanism of increased 18F-FDG uptake from the early to the delayed phase in chondroblastoma. However, Tian et al. indicated that dual-time-point 18F-FDG PET may be helpful for differentiating malignant from benign bone tumors (18F-FDG PET for such discrimination were 90.6%, 76.0%, and 83.7%, respectively, when 10% RI was considered as the cut-off point.On the other hand, there have been no reports evaluating the changes in 18F-FDG uptake, which increases from the early to the delayed phase in chondroblastoma, can result in misdiagnosis of this tumor as a malignancy.Our two presented cases would be labeled false-positive if the mentioned criteria were to be adopted. We should bear in mind that the Temporal bone chondroblastoma appears as a well-defined osteolytic mass with mild enhancement on CT images. Calcification within the lesion has been documented in 20-50% of cases . On MRI,To the best of our knowledge, the features of these tumors on diffusion-weighted images have not been reported. Based on the findings in our case report, chondroblastoma is not likely to show elevated signal intensity on diffusion-weighted images.Our two cases were preoperatively diagnosed as giant cell reparative granuloma and giant cell tumor, respectively. Osteoclast-like, multinucleated giant cells are histological common findings in chondroblastoma, giant cell reparative granuloma, and giant-cell tumors , 9, 10. In the surgical specimen, in addition to the detection of a chondroid matrix, immunohistochemical analysis of S-100 expression assisted us in making the final diagnosis of chondroblastoma. Demonstration of S-100 positivity in chondroblasts is helpful in differentiating chondroblastoma from giant cell reparative granuloma and giant cell tumors , 10.18F-FDG PET/CT findings of giant cell reparative granuloma. On the other hand, giant cell tumors are known to show markedly elevated 18F-FDG uptake (18F-FDG uptake.To the best of our knowledge, there have been no reports indicating G uptake , 8. In f18F-FDG uptake, which increases from the early to the delayed phase on PET/CT images, are encountered, chondroblastoma with its specific CT and MRI features should be considered in the differential diagnosis.In conclusion, we reported two cases of temporal bone chondroblastoma. Based on the findings, when temporal bone tumors with markedly elevated No potential conflicts of interest were disclosed.


Monomelic amyotrophy is an uncommon, benign, unilateral disorder of the lower motor neurons, affecting predominantly the hand and forearm muscles. Proximal involvement of the arm and shoulder muscles is an unusual presentation that has been rarely reported in the literature.A 28-year-old white man presented with insidious-onset, slowly progressive, unilateral weakness and atrophy of his left shoulder girdle and deltoid muscles. A neurological examination revealed weakness and atrophy in his left deltoid, infraspinatus and supraspinatus muscles. Electromyography demonstrated an active and chronic neurogenic pattern affecting his left C5 and C6 myotomes; magnetic resonance imaging of his cervical spine was normal. He did well with conservative treatment.Upper limb proximal form of monomelic amyotrophy is a rare clinical entity with a wide differential diagnosis. Physicians, especially neurologists, should be familiar with this benign condition to avoid inappropriately labeling patients as having amyotrophic lateral sclerosis and other disorders with less favorable outcomes. Hirayama tremity” . It affetremity” –5. One rWe report the case of a patient with MA, who presented with symptoms and signs in his proximal upper limb, a location rarely described in this disease –7.A 28-year-old, right-handed white man presented with insidious-onset, slowly progressive weakness and wasting of his left deltoid and shoulder girdle muscles for the last 5 years. Initially, he noticed difficulty with abducting his left arm and raising it above head level. The weakness progressed slowly over the first 3 years of initial symptoms, and then became static. He denied history of trauma, shoulder pain, or sensory symptoms, and he was not on any medications prior to development of symptoms.A neurological examination demonstrated significant atrophy of his left deltoid, supraspinatus and infraspinatus muscles, with no fasciculations Fig. . His musNerve conduction studies (NCS) of his left upper limb were normal. Electromyography (EMG) revealed fasciculations and fibrillation potentials in his biceps, deltoid and supraspinatus. Large, polyphasic motor unit action potentials with prolonged duration and reduced recruitment pattern were seen in the same muscles; the infraspinatus could not be tested due to severe degree of atrophy. EMG of other muscles in his upper and lower extremities, as well as cervical paraspinal muscles, was normal.Blood investigations, including complete blood count, sedimentation rate, renal, liver and thyroid function tests, creatine kinase (CK), and vitamins. B12 and D3 were normal. There were negative results for vasculitis screening  and viral serology: human immunodeficiency virus (HIV), hepatitis B and C. Magnetic resonance imaging (MRI) of his cervical spine, done in the neutral position, was normal  of the fingers in affected hand, exaggeration of weakness on exposure to cold (cold paresis), and absence of sensory loss, pyramidal, cerebellar, cranial nerves, and sphincter deficits , 10, 12.The differential diagnosis of MA includes the distal form of spinal muscular atrophy, amyotrophic lateral sclerosis (ALS), post-polio syndrome, multifocal motor neuropathy with conduction block (MMNCB), as well as structural lesions of the cervical cord. These clinical entities can be identified by specific clinical, radiological and electrophysiological features.et al. first proposed that a tight dural canal may be an underlying predisposing factor [The pathophysiology of MA remains unknown , but sevg factor . Hirayamg factor  suggesteMRI findings in MA are conflicting, with some studies showing localized asymmetric spinal cord atrophy, prominence and enhancement of posterior epidural venous plexus, and anterior shifting of posterior dural sac on flexion . In one An autopsy study on a patient with MA noticed anterior horn cell shrinkage and necrosis, various degrees of degeneration of small and large nerve cells, mild gliosis, and some circulatory insufficiency in the territory of the spinal cord. These abnormalities extended from C5 to T1 levels but were more marked at C7 and C8 .Electrophysiological studies (NCS and EMG) are an essential part of the diagnostic process, showing mostly chronic denervation changes in affected muscles, with or without acute denervation potentials . These studies are also helpful in excluding other apparently similar conditions . Milder denervation changes can be seen in clinically unaffected muscles as well .There is no consensus on the treatment of MA, but the use of cervical collar therapy during the acute progressive stage of the disease has been advocated . The rolMA should be suspected in patients presenting with slowly progressive weakness and atrophy restricted to one limb, followed by a static phase. While most reported cases involve the lower cervical myotomes, affecting the hand and forearm muscles, proximal upper limb involvement can be seen rarely. EMG and MRI studies are helpful in confirming the diagnosis and ruling out other clinical entities presenting in a similar fashion. Treatment is conservative in most patients, with the use of a cervical collar in appropriate cases, and physiotherapy.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 the journal.