Osteomas are benign bone tumors which arise from the cortex or medulla of craniofacial and jaw bones. They are usually asymptomatic or present as slow-growing painless masses. Larger lesions may present with aesthetic  and functional disturbances . This paper highlights a case of solitary peripheral osteoma composed of a compact bony mass arising from the lower border of the mandible in an adult female patient. The lesion presented with discomfort during deglutition, which was attributed to impingement of muscles of the oral cavity floor, including the anterior belly of digastric muscle. They commonly involve cranial and facial skeleton, including the paranasal sinuses and jaw bones. Based on their site of origin, osteomas are classified as peripheral  and central . Peripheral osteomas are more common and arise from the cortical bone, whereas central variants develop within the endosteum of bone. Rarely, osteomas can develop as extra-skeletal lesions located in soft tissues, commonly in muscles.2 Osteomas might be composed of either dense compact bone with scanty marrow spaces (compact/ivory osteoma) or cancellous bone with abundant bony trabeculae along with fibro-fatty marrow tissue (cancellous/trabecular/spongy osteoma).Osteomas invariably present as a slow-growing single bony mass (solitary lesion) with a sessile or pedunculated base. Multiple osteomas of the jaw may be present as part of Gardener syndrome along with features like intestinal polyps, multiple supernumerary/impacted teeth and tumors of soft tissues.4 This paper reports a case of PO in an adult female patient, arising from the lower border of the mandible. The bony mass presented with discomfort during swallowing due to impingement of the muscles of the oral cavity floor and the anterior belly of the digastrics muscle.Peripheral osteomas (PO) may go unnoticed when small or present as painless slow-growing bony hard masses often projecting as mushroom-shaped outgrowths from the outer cortex of the jaw. Larger mandibular lesions may cause pain, facial asymmetry, deviation of the jaw or difficulty in breathing.A 40-year-old Indian female consulted our oral and maxillofacial clinic, complaining of discomfort during swallowing. The patient was apparently alright two months back when she developed mild discomfort during deglutition. Around the same time the patient also noticed a painless swelling over the upper left side of the neck, which gradually kept increasing in size. No significant medical history or history of previous trauma was reported. On extraoral examination, no gross facial asymmetry was noticed. Intraoral examination showed upper and lower jaws with normal teeth. No other mucosal abnormality was detected over oral and oropharyngeal mucosa. Palpation of the inferior border of the mandible revealed a painless bony hard mass over the inferior border of the mandible in relation to the left canine‒premolar region. The growth was fixed to the inferior border and lingual surface of the mandible and did not show any movement during deglutition . CervicaAn orthopantomogram showed a well-defined homogenous radiopaque mass arising from basal part of the mandible below the apices of the left premolar and the first molar teeth with a continuous contour . ComputeSurgical excision of the tumor was planned and executed under general anesthesia. The surgical approach used was submandibular incision placed in the upper neck crease. Upon layered dissection over the lower border of the mandible, the anterior belly of the digastric muscle was seen overriding the lesion on its buccal aspect . The mus5 Based on its site of origin they can be classified as central (arising from the medullary cavity) or peripheral (arising from the cortex). Osteomas in the head and neck region frequently involve paranasal sinuses and frontal bone. Other bones involved include the temporal bone, orbit, external auditory canal and pterygoid process. PO of the jaw skeleton is more commonly seen in the mandible as compared to maxilla. Sayan et al in a review of 35 PO cases found that the majority of the lesions involved the frontal bone (28.57%), with the mandible and maxilla accounting for 22.85% and 14.28% of cases, respectively.6 Mandibular PO occurs predominantly in the body‒ramus region and condyle. Those arising from the dentate segment may involve the buccal/lingual cortex or the lower border of the mandible. In the present case the lesion developed from the basal bone of the mandible in the premolar molar region. Cases of PO arising from the coronoid process and sigmoid notch have also been reported in the literature.7Osteomas are defined as benign osteogenic lesions predominantly composed of osteoblastic connective tissue which forms abundant osteoid and new bone that eventually mature to compact bone over a period of time.2 Osteomas show a slow continuous increase in size without growth cessation at adulthood. This characteristic feature helps distinguish osteomas from other bony exostoses and supports a neoplastic origin. In the present case no history of trauma or any overt cause of chronic irritation could be identified, possibly suggesting neoplastic etiology.The pathogenic process of osteoma is not clearly understood. Some authors classify it as a true neoplasm while others consider it as a reactive lesion secondary to chronic irritation or trauma.4 Depending upon its location, mandibular PO may cause other functional disturbances like occlusal dysfunction, pain, sensory deficit and ulceration of the overlying mucosa. Condylar osteomas produce limited mouth opening and mandibular deviation. A case of large PO of the mandible causing facial asymmetry, deviation of the tongue and respiratory obstruction has been previously reported.8 None of the previous cases of PO of the mandible have been reported to present with difficulty swallowing as in this report. CT imaging showed the lesion impinging on the muscles of oral cavity floor. Surgical excision is the treatment of choice for PO. Recurrence is relatively infrequent and malignant transformation has not been reported.Clinically, mandibular PO presents as an asymptomatic slow-growing bony hard mass which may be diagnosed during routine radiographic examinations. As the lesion grows in size, it causes an intraoral  swelling or an extraoral mass causing facial asymmetry.Presentation of PO depends upon the size and location of the pathology. The majority of the cases are asymptomatic or presents as a slow-growing bone mass. Larger lesions may cause intraoral or extraoral swelling with facial asymmetry. Other less frequently reported symptoms include difficulty in breathing, deviation of the tongue and ulceration of the superficial mucosa. This paper highlights a rare presentation of PO of the mandible causing difficulty during deglutition due to impingement of muscles of the oral cavity floor.The authors would like to thank Dr. Ajay Nayak, associate professor, Department of Oral Medicine and Radiology, School of Dental Sciences, Karad, India.KN preformed the surgical procedure. KN and SV were involved in diagnosis and formatting the case report. AV drafted the manuscript and all the authors contributed to the revision and final approval of the manuscript.The authors report no funding for this article.The authors declare no competing interests with regards to the authorship and/or publication of this article.The authors declare that the patient, whose data is reported in this article, has given written consent to the authors and the Ethics Committee of Krishna Institute of Medical Sciences Deemed University, Karad, India for the publication of this paper.


Magnetic resonance imaging (MRI) of the lumbar spine showed a multilevel high intensity epidural mass lesion compressing the dural sac extending from L1 to L5 level, on T1 and T2 weighted images (WI) (A & B). Axial MRI T2-WI showing a hyperintense anterior epidural mass with deformation of the dural sac at the level of L4 (C). Frontal MRI myelography showed a complete stop of the cerebro-spinal fluid at lumber levels (D). The patient was operated. Laminectomy and epidural decompression at L3, L4 and L5 levels with removal of epidural fat has been performed. Pathologic examination revealed nodular proliferation of mature fat cells, with lipomatosis. Postoperative examination showed improvement of clinical signs.A 50-year-old male without medical history presented with progressive development of motor weakness in his lower extremities and urinary leakage. For one year, he had experienced severe lower back pain associated to bilateral radicular claudication of the lower limbs. Physical examination revealed stepping with bilateral foot dorsiflexor weakness (2/5). Deep-tendon reflexes were abolished in both lower limbs with hypoesthesia of the lower left extremity. Examination of Upper limbs was normal and the body mass index was 37 kg/m


Chondroma is a benign tumour of mesenchymal origin that is composed of cartilage and rarely located in soft tissues, being described so far only in four cases, as located in the bladder, according to our knowledge. We describe the fifth case of a 67-year-old woman who consulted for microscopic haematuria, with an endoscopic finding of submucosal nodular image in the anterior wall of the bladder, which after resection and the histologic study shows cartilage and fibroconnective tissue, in part hyalinised, and positive immunohistochemical staining of cells with vimentin and S-100; this fact can support the diagnosis of bladder chondroma. A 67-year-old woman with a history of hypertension, dyslipidemia, hypothyroidism, osteoporosis, and mixed anxiety-depressive disorder, as well as a history of mastectomy plus axillary lymphadenectomy for breast cancer 16 years ago and in remission, is referred to the Urology Department for persistent microscopic haematuria.The patient denied having any abdominal pain, dysuria, frequency, tenesmus, or some other urinary symptoms. The urinalysis confirms the presence of persistent microscopic haematuria of between 60 and 100 red blood cells per field, and a urine culture is performed which comes back negative. Urologic ultrasound was requested, which showed the presence of an avascular echogenic image with a polypoid aspect of approximately 16 × 20 mm in the anterior wall of the bladder, so that endoscopic examination is performed under anaesthesia, evidencing raising or elevation of the bladder mucosa, with a cystic appearance on the anterior wall of the bladder of approximately 15 mm, which is completely resected with a resection loop for subsequent pathological analysis. The recovery of the patient was uneventful.Macroscopically, two white-brown fragments of bladder wall with muscular layer were analysed. The fragments together measure 14 × 7 × 4 mm. Microscopically, one of the fragments corresponds to fibroconnective tissue compressed by nodular and lobulated tumour composed of hyaline cartilaginous, without cellular atypia and with basophilic areas Figures .Immunohistochemical study of the tumoural tissue was immunostain positive for S-100  and vimeIn two years of follow-up, the patient remained asymptomatic, with negative urine cultures, normal cystoscopy with previous resection scar on the anterior wall without pathological findings, and urologic ultrasound within normal limits.Chondroma is a benign tumour of mesenchymal origin and slow growth that is composed of cartilage and is rarely located in soft tissues, being more commonly described in the fingers, limbs, and head or neck . To dateOur case, as in the previous four, has been in women with an age between the fifth and seventh decade of life. Three of the previously described patients presented some type of abdominal pain at the time of diagnosis, whereas, in the present case, it was an incidental finding during the study of microscopic haematuria, very similar to the last case described in the literature. The endoscopic image seems to coincide in the 5 cases, since it is a smooth submucosal tumour and located in the anterior bladder wall.Soft tissue chondromas usually affect both sexes equally, between 40 and 70 years of age . The oriSome studies seem to correlate the pathogenesis of this tumour with the possible genetic influence demonstrated in monosomies, trisomies, translocations, and rearrangement of trisomy 11 in cytogenetic studies of soft tissue chondromas ; howeverFrom the histopathological point of view, the findings can be variable and may exist from mature forms with hyaline cartilage arranged in lobes  to immature forms with preponderance of chondroblasts . Like thThe main differential diagnosis to be taken into account is chondrosarcoma, which presents a malignant course and can mean a great challenge for the pathologist to be able to differentiate a low-grade chondrosarcoma from a soft tissue chondroma , 3.Therefore, soft tissue chondroma in the bladder is a possible pathology, although rare, and every pathologist should think of it as a possible entity in cases of masses with presence of subepithelial hyaline tissue.It is necessary to accumulate more cases to obtain adequate conclusions and to better understand the causes of this pathology; however we can say that bladder chondroma is presumably an extremely rare condition and benign behaviour and must be taken into account during the pathological analysis in order to reach its possible diagnosis.


An 18-months-old male infant presented with a rapidly growing tumor on the right upper eyelid. Orbital computed tomography (CT) revealed a large, well-circumscribed mass with low density signal in the right upper eyelid. Magnetic resonance images (MRI) showed a lesion of mixed T1-signal intensity and high signal intensity in T2-weighted images. The tumor was treated by simple anterior orbitotomy with excisional biopsy, and the diagnosis of fibrolipoma was made by histopathologic examination. There was no evidence of tumor at the four-year follow-up. Fibrolipoma is one of the rare variant of the lipoma and only four cases have been reported in the orbit including the present case. Except for this case all other cases were reported in adults. Lipomas are common benign soft tissue neoplasms of mature adipose tissue , 2]. Al. Al2]. AAn 18-months-old male developed a tumor on his right upper eyelid over the course of two months. The tumor became significantly prominent, progressive enlargement over the last month. There was history of trauma to the area. On physical examination he was found to have a very large tumor, approximately 2×2 cm on the right upper eyelid, there appeared to be no discolorations, erythema, or signs of inflammation, it was a firm mass, and nontender to the palpation Figure 1 . The remLipomas are well-circumscribed slow growing, benign tumors composed of mature fat cell grouped in lobules by connective tissue septa . These tFibrolipomas are more heterogeneous than lipomas on MRI images. On CT images, lipomas can be seen as an uncontrasted hypodense mass .The exact etiology of lipomas and their variants is not well established. An association with genetic aberrations has been described for most types of adipocytic tumor and some trauma mechanisms, such as origin from lipoblastic embryonic cell nest, blunt trauma or chronic intermittent compression, have been associated with the apparition of lipomas . Fatty dBecause of the diverse modes of presentation, some other lesions should be considered in the clinical differential diagnosis and these include fibrous histiocytoma, fasciitis nodular and schawannoma. Definitive diagnosis can be based on histological appearance. The treatment of fibrolipoma, including all the histological variants is complete surgical excision. In our case the patient requested the removal of his lesion because of concerns rapidly growing, and the esthetic even so functional aspects resulting from the compression of focal structures. The most common symptom described was rapidly growing painless swelling. Medical management of lipomas includes steroid injections (triamcinolone acetonide) that result in local fat atrophy, thus, shrinking the tumor size. They are best done on lipomas that are less than 2.54 cm in diameter. Average volume of steroid used may range from 1 to 3 ml depending on the size of tumor . In summThe authors declare that they have no competing interests.


Inguinal hernia is one of the most common benign pathologies that primarily affects men. Primary gastrointestinal non-Hodgkin’s lymphoma (PGI NHL) is the most common type of extranodal lymphoma. This study reports a rare case in which these two conditions co-exist.An 85-year-old male complained of bowel movement pattern change, abdominal distension and loss of weight, without vomiting but with nausea. A computed tomographic scan of the abdomen showed a small bowel obstruction caused by a migration of a small bowel loop in the right inguinal canal, with a clinically non-reducible inguinal hernia. The patient underwent surgery. The histopathological report showed small bowel large B cell non-Hodgkin’s lymphoma.When the diagnosis of the contents of an inguinal hernia is not well-established, surgery should be performed as soon as possible to ensure the cure of the disease and the correct diagnosis of the contents. Inguinal hernia is one of the most common benign pathologies that primarily affects men; surgical hernia repair procedures account for approximately 2 million surgical cases annually worldwide, with 850,000 of those performed in the USA .The small bowel  is the sHere, we report a rare case in which these two conditions co-exist and their treatment.bowel movement pattern change, abdominal distension and loss of weight, without vomiting but with nausea. Clinical examination revealed abdominal distension (Fig. 3, and his neutrophil count was 96.9%. A computed tomographic scan of the abdomen showed a small bowel obstruction caused by a migration of a small bowel loop in the right inguinal canal, with full-thickness expansion of the bowel wall (Fig. An 85-year-old male was admitted to the hospital complaining of an approximate 1 month of all Fig. . The patOn exploration, fluid was found in the peritoneal cavity, and the ileal loop was lodged within the interior inguinal ring. A combined inguinal approach was deemed necessary. On exploration, a small bowel mass firmly adherent to and inseparable from the testicle was found. A bowel resection including the mass and the testicle was performed Fig. . An ileoApproximately, 10% of inguinal hernias become irreducible; this is often due to benign factors, whereas uncommonly, co-existing malignancy can be the cause of the irreducibility .Malignant masses in inguinal hernias appear in less than 0.5% of excised sacs .Tumours in the hernia sac can be classified based on Lejar’s studies as saccular and intrasaccular: saccular when they are primary, such as mesothelioma or when metastatic disease involves the peritoneal sac; intrasaccular when the sac contains an organ affected by a malignant tumour .The majority of intrasaccular tumours are of intestinal origin, and the first case that was reported was a sigmoid colon carcinoma .The sigmoid colon is involved in most cases; however, reported cases do not exceed a maximum of four cases per author. The most recent review reported a total of 27 colon carcinoma cases within inguinal hernia sacs and represents most of these patients present in the literature .Other tumours of the intestine are rarely encountered, and they usually originate from the small bowel; eight such tumours have been reported to date , 9–15.Other types of intrasaccular tumours, less frequent than the tumours of intestinal origin, include ovarian and bladder cancer tumours. Ovarian tumours presenting as an inguinal hernia have been described in ten cases, with patients ranging in age from 43 to 81 years . InguinaLymphoma diagnosed as an inguinal/femoral hernia is described in a few cases. A review of David R. and colleagues  reportedOnly one case in the literature is reported as small intestine lymphoma diagnosed as a femoral hernia , and oneIn conclusion, this case report suggests that when the diagnosis of the contents of an inguinal hernia is not well established, surgery should be performed as soon as possible to ensure that the disease is cured and that the correct diagnosis of the contents is determined. In cases of a curable tumour, the lapse of time may be detrimental to the patient.


Here, we describe the first reported case of osteosarcoma occurring postsurgically in a tracheotomy tract, highlighting the possibility of osteosarcoma seeding during head and neck surgery. Preventative measures such as performing the tracheostomy after the tumor resection while walling off the tracheostomy site from the operative field should be considered. Osteosarcoma is the most common primary malignant bone tumor in children and adolescents. However, it rarely occurs in the head and neck region, accounting for less than 1% of head and neck malignancies A 21‐year‐old women was referred to our center due to recurrent mandibular osteosarcoma, 6 months after total tumor extirpation and postoperative radiation. She also was noted to have a lump at her central neck, at the scar of the tracheotomy incision, which was performed as a part of her last operation. The woman is a p53 mutation carrier, with a family history of a father who died from mandibular osteosarcoma. At 18 months of age, she was diagnosed with a right popliteal fossa rhabdomyosarcoma for which she underwent several surgeries and VACA‐IV chemotherapy for 18 months followed by radiation. At the age of 14 years, she was diagnosed with a postradiation osteosarcoma in her right tibia. She was treated with chemotherapy, followed by amputation with intercalary allograft implantation, a debridement after graft failure, and an anterior lateral thigh (ALT) free flap reconstruction (from her left leg). Further local osteosarcoma recurrences led to an above‐knee amputation of her right leg at the age of 15. At the age of 20, she noticed a gradually enlarging, symptomatic mass in her right mandible. A biopsy was taken, which revealed an osteoblastic osteosarcoma. At that time, she had a 3‐cm exophytic lesion in the right mandible, involving the floor of the mouth, extending under the mandible. The patient underwent a right segmental mandibulectomy, selective neck dissection levels 1‐3, and tracheotomy, with chimeric scapular and titanium plate flap reconstruction at another center. The histopathologic finding was a 13‐mm high‐grade osteosarcoma, involving the mandibular bone, gingival mucosa, and the masseter muscle. The anterior and posterior margins were free of tumor. One level 2 cervical lymph node, of 18 resected, showed metastatic tumor. Because of toxicities of her previous treatments with cisplatin, doxorubicin, etoposide (VP16), and ifosfamide, she received second‐line gemcitabine and docetaxel and eventually 2 cycles of methotrexate with good response according to the PET‐CT. Two months after surgery, treatment with pembrolizumab (Keytruda), an anti‐PD‐1 antibody, was initiated. However, swelling in her right mandible recurred. In addition, a lump in her central neck, at the site of the tracheostomy scar, was noted  Planning the operation carefully by first removing the primary lesion, and only after tumor removal, irrigation and site isolation, continuing toward and neck dissection and/or tracheotomy. (2) Meticulous irrigations with hypotonic saline solution after primary lesion removal. (3) Creating a mechanical barrier by covering the tracheotomy site and the primary lesion site, with a nonpenetrating wound dressing. (4) Changing gowns, gloves, and instruments after the primary lesion removal and before performing tracheotomy, reconstruction, and closure. (5) Consider including the entire operative field in the postoperative radiation field when postoperative radiation therapy is indicated.The case we present is not the usual seeding in the tract of a biopsy. To the best of our knowledge, this is the first case reported of iatrogenic seeding of head and neck osteosarcoma. Also, this is the first reported case of an osteosarcoma occurring in the tract of a tracheotomy that was carried out during mandibulectomy for mandibular osteosarcoma. This report highlights the possibility of osteosarcoma metastases occurring by direct implantation during head and neck surgery. Consequently, careful consideration should be given to possible preventative measures such as performing the tracheostomy after resection of the tumor and/or walling off the tracheostomy site from the operative field during resection of head and neck osteosarcoma.ID: wrote manuscript, conception and design, participated in patient's management, operation, and follow‐up. RL: involved in drafting the manuscript, revising it critically for important intellectual content. SA: reviewed the pathological aspect of the case, examined the specimens, provided pathological diagnosis and histopathologic figures for the manuscript. ZG: managed the case, from first diagnosis and decision‐making, to planning and performing the operation and patient's follow‐up.None of the authors has any conflicts of interest to disclose.


Odontogenic fibroma (OF) is considered an unusual, benign mesenchymal neoplasm and one of the most little-understood lesions amongst all odontogenic neoplasms. The incidence rate of the tumor is from 0% to 5.5%. WHO classified them into intraosseous or central and extraosseous or peripheral variants. It chiefly consists of fibroblastic tissue with an inconsistent amount of inactive appearing odontogenic epithelium. The lesion has a slow growth along with cortical expansion with equal predilection in the anterior maxilla and posterior mandible. Radiologically, multilocular radiolucency is the most frequent finding with few cases being associated with root resorption or displacement. Microscopically, mature collagen fibers and numerous fibroblasts along with odontogenic epithelial islands are characteristically found. Central Odontogenic Fibroma responds well to surgical enucleation with no tendency for malignancy or recurrence. Here we report a rare case report of an 18 yr old male patient with Odontogenic fibroma of the posterior maxilla. The Odontogenic fibroma (OF) is an unusual, atypical, benign tumour with defined margins and of mesodermal origin . It is aCentral odontogenic fibroma (COF) is benign and distinctive variant, small or extensive, probably has finite growth. Extensive bone destruction and expansion of bone indicate the aggressive nature of the lesion. Thus, not many cases of this unusual lesion have been recorded and published. According to a literature, only 7 cases have been reported in the last 5 to 6 yr tenure. This paper reports a case of odontogenic fibroma in the posterior maxillary region of an 18 yr old male patient.An 18 yr old male patient presented with pain in the left upper posterior region of the jaw since 2 months. The pain was dull and intermittent in nature that aggravated on mastication or pressure and relieved on taking medication. It was occasionally associated with pus discharge. Past dental history revealed that he had undergone extraction of upper left posterior tooth due to caries. The patient also reported with good systemic health. There were no conflicts of interest and no financial issues regarding this case.There was no facial asymmetry seen extra orally. On intraoral examination, no significant swelling or vestibular changes were evident. Clinically, 27 and 28 were missing.  The alveThe intraoral periapical radiograph showed irregular radiopaque masses distal to 26 in the alveolar bone. OPG showed a well-defined radiolucency present in the 27, 28 regions that extended from distal of 26 to maxillary tuberosity region surrounded by a white sclerotic border with intermittent irregular radiopaque masses within. Vertically impacted 28 were seen embedded in the left zygoma region.  based onOn histopathological examination, the H & E section revealed a highly fibrocellular connective tissue stroma consisting of numerous strands of odontogenic epithelial islands composed of peripherally arranged tall columnar cells. The stroma consists of collagenous bundles with plump shaped fibroblasts and endothelial lined blood vessels with extravasated RBCs. Deeper section showed the presence of few basophilic calcified areas . ConsideCOF represents about 9.4% of all odontogenic tumours. The most common odontogenic tumour is the odontoma with OF occupying the fourth highest position in order of tumour frequency . OF havePresently, WHO type (lesion with dysplastic hard matrix) should be termed as OF complex type or fibroblastic OF . ClinicaRadiologically, it presents as uni- or multilocular radiolucency with distinct borders. In very few cases, it could present with a mixed radiolucent and radiopaque appearance ill-defined borders. Root resorption and displacement of teeth have been reported in cases of more severe lesions . ApproxiMicroscopically, the lesion is characterized principally by dense mature collagen intermingled with fibroblasts and fibrocytes. The collagenous tissue being reasonably dense to dense in most areas and inactive odontogenic epithelium in strands or nests may be present . WHO cenAs treatment, enucleation and curettage produce sufficiently favorable results. COF does not have a defined capsule, but it has limited potential for growth. The treatment is conventional surgery with minimal invasion through the enucleation of the neoplasm followed by the curettage of the cavity to enhance proper healing. Recurrences are not common. Among the cases reported in literature, few have shown recurrence. Thus, this pathology has a sufficiently favorable prognosis , 8.COF is an infrequent benign mesenchymal neoplasm with several subtypes originating due to several reasons that facilitate its pathogenesis and its subtypes. The importance of periodic clinical and radiographic examinations demonstrated for the timely detection of this kind of pathology. Moreover, it demonstrates the importance of adequate experience in the treatment and post-operative follow-up to detect the rare occurrence of the lesion.The authors declare that there is no Conflict of Interests.


Abdominal apoplexy is a rare and fatal emergency event, which is coined as a comparison to the cerebrovascular apoplexy. The exact mechanism of abdominal apoplexy was unclear, but arteriosclerosis, hypertension, abdominal aneurysm, and other predisposing angiopathy were considered to be the main reasons of abdominal apoplexy. The development of the imaging technology gave us more opportunities to confirm the diagnosis of abdominal apoplexy. However, the diagnosis and identification of the bleeding sites still continued to be a challenge.A 55-year-old man presented to the emergency department with chief complains of sudden severe abdominal pain.The patient was diagnosed as abdominal apoplexy with 2 synchronous bleeding sites.Angiography confirmed diagnosis of abdominal apoplexy and revealed 2 synchronous bleeding sites in gastroduodenal artery (GDA) and inferior pancreaticoduodenal artery (IPDA). Transcatheter embolization was performed immediately.The patient recovered and was discharged very soon. Two months later, the patient totally recovered and the hematoma disappeared in the CT imaging.The reported case is rare, given the very low incidence of abdominal apoplexy with 2 synchronous bleeding sites in GDA and IPDA. The awareness of abdominal apoplexy was still the key point in the management of this disease. Quick diagnosis by the imaging and immediate embolization were very important for the treatment. However, the diagnosis continued to be delayed because of rarity of the process and lack of suspicion. The preoperative diagnosis remains challenging, despite the recent radiologic advances and surgical exploration is often unavoidable for the diagnosis and treatment.Abdominal apoplexy is a rare and fatal emergency event, which was replaced by “idiopathic spontaneous intraperitoneal hemorrhage (ISIH).” The term abdominal apoplexy is still widely used, because it is coined as a comparison to the cerebrovascular apoplexy. Although not completely understood, there may be the similar feature and etiology between abdominal apoplexy and cerebrovascular apoplexy. Historically, the diagnosis of abdominal apoplexy and the site of bleeding could only be confirmed during the surgical intervention or autopsy, and eventually around 30% of cases had no identifiable source.Usually the abdominal apoplexy had only 1 identified bleeding site, but we reported a case presenting with 2 synchronous bleeding sites, involving gastroduodenal artery (GDA) and inferior pancreaticoduodenal artery (IPDA).2A 55-year-old man presented to the emergency department (ED) with chief complains of abdominal pain. He suffered sudden upper abdominal pain with nausea and vomiting. The pain was severe and the patient had to keep a curled up posture to relieve the pain. He had progression of his pain through to the back and subjective sensation of abdominal distention. His past medical history included hypertension, hyperlipidemia, and cerebral infraction. He suffered ischemic cerebral infraction 4 years ago and recovered without limb dyskinesia. He had no other antecedent history. He denied use of tobacco or alcohol. There were no medication changes recently. Review of systems was generally unremarkable, and he specifically denied stool changes or other associations. He denied any trauma. Vital signs were remarkable for a blood pressure of 116/70 mm Hg with a heart rate of 92 beats/min. He had upper abdominal tenderness with absence of bowel sounds. His blood pressure remained stable. Hemoglobin was 10.2 g/dL with a hematocrit of 37.5%. Serum amylase was elevated at 150 U/L . Serum lipase was elevated at 696 . The other chemistry studies were essentially normal. Coagulation studies were all within the normal range. Due to the patient's body habitus and bowel gas, there were no positive signs in sonography. Considering the feature of abdominal pain with radiation to the back and the history of cerebrovascular apoplexy, the visceral vascular disease was highly suspected. Because the patient remained hemodynamically stable, he was sent for an abdominal enhanced computed tomography (CT) scan. The enhanced CT scan was negative for abdominal aneurysm, but showed a 12 cm mass with extravasation of the contrast medium  and atherosclerosis (80–87%). So besides visceral artery aneurysms, the hypertension and atherosclerosis might be also important reasons for the abdominal apoplexy. Of course the mismatching could also be caused by the high proportion of cases without identifiable bleeding sites and the respective rupture risks of different aneurysms. Arteriosclerosis also was one of the reasons for the aneurysm, especially dissecting aneurysms. It was generally believed that the most common cause of abdominal apoplexy in older patients was arteriosclerosis with or without hypertension. In younger patients congenital defects or systemic disease might account for the hemorrhage such as polyarteritis nodosa and Ehlers-Danlos syndrome.,12,13Historically, abdominal aneurysm was considered to be the main reason of abdominal apoplexy. Of all visceral artery aneurysms, 60% involve the splenic artery, 20% hepatic artery, 5% superior mesenteric artery, and 3% gastric artery and gastroepiploic arteries. Carter reviewed 2 cases of GDA and 2 cases of IPDA in 85 cases of abdominal apoplexy (35 cases undetermined). Kleinsasser reviewed 3 cases of GDA and 3 cases of IPDA in 83 cases of abdominal apoplexy (23 cases undetermined). The rupture of GDA usually was caused by artery dissections, but GDA dissections were particularly rare.–16 The anatomic area of GDA may be susceptible to mechanical stress contributing to small artery dissection in combination with other poorly defined predisposing conditions. The aneurysm of IPDA was also rare and usually caused by atherosclerosis, pancreatitis, and trauma with the symptoms of gastrointestinal or retroperitoneal hemorrhage.–21There were 2 arteries involved in our case: GDA and IPDA. To our knowledge, this is the first reported case of abdominal apoplexy that has 2 identified bleeding sites. Historically, about 70% cases of abdominal apoplexy had identifiable source of bleeding, and there was only 1 bleeding site in each identifiable case. Both GDA and IPDA were the rare sources of hemorrhage. Kennedy reported 2 cases of GDA and 1 case of IPDA in 50 cases of abdominal apoplexy (19 cases undetermined). We believed that there might be some equivalence between multifocal cerebrovascular apoplexy  and multifocal abdominal apoplexy. Without the pathological evidence, the exact etiology of our case was undetermined. But the angiography revealed the absence of obvious aneurysm and the bleeding sites located at the branches of 2 arteries. Considering the history of hypertension and cerebrovascular disease, the possible mechanism in our case might be atherosclerosis, hypertension or multiple microaneurysms. Also, 1 of the 2 arteries might be the primary site of bleeding, and the other was secondary to the traction of retroperitoneal hematoma caused by the primary bleeding.The abdominal apoplexy with 2 bleeding arteries was extremely rare, considering the low incidence of each artery in the literature. However, cerebrovascular apoplexy or atraumatic multifocal intracerebral hemorrhage was also extremely rare but had been described in the literature. Multiple contributing factors have been correlated to multifocal intracerebral hemorrhage in the literature, which included hypertension, multiple microbleeding, cerebral angiopathy , vasculitis, and so on. In our case, the enhanced CT revealed extravasation of the contrast medium and the diagnosis of abdominal apoplexy was confirmed. Then the subsequent angiography confirmed the bleeding sites and successful transcatheter embolization was performed with satisfied prognosis. Spontaneous stop of bleeding from GDA or IPDA had been described in the literature,,23 but this was extremely rare, so the active treatment was widely recommended.In the emergency situation of sudden abdominal pain and undetermined hypotension, the awareness of abdominal apoplexy was still the key point in the management of this unexpected catastrophic attack. Diagnostic evaluation specific to abdominal apoplexy was largely dependent on imaging techniques. CT angiogram was very useful in making the diagnosis, localizing the bleeding vessel, and guiding subsequent surgical and angiographic management.,4 But if the patient was hemodynamically unstable with undetermined diagnosis from the CT angiogram, exploratory surgery should be employed. Unfortunately nearly 30% to 40% surgeries could not localize the site of bleeding or just find hematoma of mesentery even through the laparotomy, and reported mortality with nontherapeutic exploratory laparotomy varies from 40% to 66%. The reason for nontherapeutic exploratory laparotomy might include the lower pressure and the site of bleeding. The rupture of GDA or IPDA usually caused hematoma in the retro peritoneum, around the head of pancreas and duodenum. Surgical exploration of this area might be difficult owing to the multiple small vessels from the pancreaticoduodenal arcade and the unexpected risk of damage to the pancreas and duodenum. In the condition of our case, embolism might be a better choice than laparotomy.Historically, the nonsurgical mortality had approached 100% and laparotomy was recommended as the first therapy for the abdominal apoplexy. However, the development of imaging technology gave us more opportunities to diagnose the abdominal apoplexy and localize the suspected bleeding vessels, which might somewhat change the treatment strategy of abdominal apoplexy in selected patients. Now embolization was much preferred because of less invasion if the patient was hemodynamically stable.4Abdominal apoplexy is a rare and fatal emergency event. The exact mechanism of abdominal apoplexy was unclear, but arteriosclerosis, hypertension, abdominal aneurysm, and other predisposing angiopathy were considered to be the main reasons of abdominal apoplexy. The development of the imaging technology gave us more opportunities to suspect or confirm the diagnosis of abdominal apoplexy. The awareness of abdominal apoplexy was still the key point in the management of this disease. Quick diagnosis by the imaging and immediate embolization were very important for the treatment.


Primary non-Hodgkin’s lymphoma (NHL) of the spine is a rare form of extranodal lymphoma. This entity constitutes a diagnostic challenge due to its mimicking of other spinal diseases and the difficulty in establishing a tissue diagnosis. In fact, core biopsy can be inconclusive, oftentimes requiring surgical biopsy. Definitive evidence is lacking regarding the treatment of choice. As a result, the prognosis remains unfavorable. We present the case of an adult female who presented with back pain and was found to have a spinal NHL. We also review the literature regarding this rare occurrence. Primary spinal lymphoma is a rare form of extranodal lymphoma. It is thought to arise from the paraspinal lymphoid tissue and subsequently invade the spinal cord. The diagnosis of this entity is challenging due to its atypical clinical presentation and the difficulty in establishing a conclusive tissue diagnosis with core biopsy. The treatment of choice has yet to be defined, and the role of surgery remains controversial. Therefore, the prognosis of this entity remains unfavorable. We present the case of an adult female who presented with back pain and was found to have a spinal lymphoma. We also review the literature regarding this rare occurrence.A 69-year-old Caucasian female with a past medical history of hypertension, rheumatoid arthritis, Sjogren syndrome, and esophagitis presented to our emergency department for evaluation of numbness in her arms and legs. Over the last 6 weeks, she has been experiencing intermittent chest tightness and mid-back pain. Twenty-four hours prior to presentation, she developed constant moderate to severe bilateral paresthesia in her upper and lower extremities. She denied fever, night sweats, shortness of breath, palpitations, recent weight loss or weakness in her extremities.Six weeks prior to presentation to our facility, the patient had an unrevealing workup for cardiac etiologies of her chest pain at a neighboring hospital. A chest computed tomography (CT) had shown spinal sclerosis from T4 to T7, which was thought to be the etiology of her back pain. A course of non-steroidal anti-inflammatory drug provided partial relief. One month later, she noted worsening of her symptoms. A thoracic magnetic resonance imaging (MRI) with contrast had shown a 4.5 × 1.6 cm fusiform mass in the epidural space from T5 to T7, along with large bilateral paraspinal masses extending from T4 to T9 with invasion into the neural foramina. Two CT-guided biopsies of the spine had been attempted but did not yield enough tissue, and were therefore inconclusive.The patient’s surgical history included an appendectomy, bilateral total knee replacements, a hysterectomy for uterine fibroids and a breast lumpectomy in the remote past for breast cancer from which she is in remission. The patient has a documented allergy to penicillin, codeine and sulfa drugs. She stopped smoking 20 years ago and did not abuse alcohol or recreational drugs. She was independent in her activities of daily living. The patient’s medication consisted of alendronate, amlodipine, omeprazole, hydrochlorothiazide, vitamin D, methotrexate, pregabalin, calcium, pyridoxine, and vitamin B12. She was previously on adalimumab for her rheumatoid arthritis, which was stopped since the differential diagnosis of her suspicious spinal mass included extrapulmonary tuberculosis.On physical examination, the patient appeared uncomfortable due to her back pain. She had mildly decreased breath sounds on auscultation with regular heart rate and normal cardiac sounds. Her abdomen was soft and non-tender. There were no palpable cervical or axillary lymph nodes. Neurologic exam was within normal limits, with preserved motor functions, normal reflexes and no noted sensory deficits.At our hospital, since she was complaining of chest pain, a CT of the chest with contrast, intended to rule out pulmonary embolism, showed bilateral pleural effusions and an increased soft tissue density in the bilateral paraspinal compartment ranging from T4 to T8, which was suspicious for a malignant process. No endobronchial obstruction or pulmonary mass were found. An MRI of the thoracic spine with contrast subsequently revealed multiple foci of bone marrow signal abnormalities suspicious for malignancy, and infiltration of the T4 to T6 vertebral bodies. Extensions into the neural foramens, spinal canal, adjacent ribs, epidural and paraspinal soft tissue were also noted on multiple levels. Additional osseous lesions were found at T7, T8, and T11. Cord compression was pronounced at the level of T4-T5. The tumor had overall progressed and increased in size compared to the prior MRI study done at another facility .The patient was diagnosed as having cord compression syndrome and promptly received high-dose steroids and pain control medications, after consultation with neurosurgery. She noted that these conservative measures improved her paresthesia. Given a high suspicious of a malignant process, her oncologist performed an extensive workup. An MRI of the brain did not show any intraparenchymal lesions. A CT scan of the abdomen and pelvis revealed no enlarged lymph nodes, and was otherwise normal. A bone marrow biopsy was normocellular with trilineage hematopoesis. Significant blood tests results are shown in In situ hybridization showed a mixed population of kappa and lambda light chain positive plasma cells. The overall pattern was consistent with a diffuse large B cell lymphoma of non-germinal center cell of origin due to MUM-1 positivity and CD-10 negativity. The CD4 to CD8 ratio by flow cytometry was 3.9 to 1. The remainder of the patient’s hospital course was unremarkable. She was discharged home 5 days later with instructions to follow-up with her oncologist in the clinic.In light of her neurological symptoms due to the cord compression and since multiple CT-guided biopsies did not yield a diagnosis, the patient underwent neurosurgical decompressive laminectomies from T4 to T8, with resection of the epidural spinal cord tumors. Significant neurological invasion limited the extent of tumor resection. Analysis of the pathological sections showed a diffuse infiltrate of lymphoid cells consistent mainly of small lymphocytes with significant number of scattered large lymphoid cells with irregular often prominent nucleoli, vesicular chromatin and abundant clear cytoplasm. Most cells were positive for the B cell antigens, CD-20 and PAX-5. The atypical large cells were strongly positive for the B cell antigen CD-20, the germinal cell antigen BCL-6 and the post germinal B-cell antigen MUM-1. BCL-2 and BCL-1 (cyclin D1) were negative as well as CD-10, CD-15, CD-21, CD-23, CD-138 and CD-30. The P-53 regulatory protein was rarely expressed. No Reed Sternberg cells were found. After discussion with the patient and her family, she was started at our ambulatory oncology clinic on an R-CHOP  regimen and intra-thecal methotrexate. A follow-up MRI after three cycles of chemotherapy showed an almost complete resolution of the paraspinal metastatic disease and epidural soft tissue extension . The patExtranodal lymphomas constitute 10-20% of all lymphomas . It usuaIn our case, malignancy was high on the differential diagnosis for a number of reasons. First, given the patient’s history of treated breast cancer, a metastatic spinal recurrence was initially highly suspected. Secondly, her history of Sjogren’s syndrome is a known significant risk factor for lymphoma. Third, treatment with adalimumab could also have precipitated the occurrence of lymphoma.Non-Hodgkin’s lymphoma (NHL) accounts for 85% of spinal lymphoma cases, with the majority being diffuse large B cell lymphomas. These primary tumors are most commonly located in the thoracic spine, followed by the cervical spine and less commonly in the lumbar spine . The preThe tumorigenesis of spinal lymphoma is not completely understood. It is thought to arise from the paraspinal soft tissues such as the paravertebral ganglions or the epidural lymphoid tissues . It is tWhen faced with a patient with a suspected lymphoma of the spine, it is first important to rule out other localizations of lymphoma with imaging, including positron emission tomography/computed tomography (PET/CT), in order to differentiate between primary spinal lymphoma and a secondary metastatic spread of a systemic lymphoma. The initial workup is similar to that of nodal lymphoma. Lack of bone destruction or erosion on CT or X-ray suggests the diagnosis of spinal lymphoma . It is, Treatment modalities lack robust evidence and involve chemotherapy, steroids, radiation therapy, stem cell transplant and surgery. As seen in our patient, a rapid neurological improvement following steroid administration is often seen, and is explained by lymphoma’s corticosteroid’s sensitivity. The literature offers conflicting evidence on the role of primary surgical decompression, in light of the tumor’s sensitivity to radiation and chemotherapy. For instance, Chang et al found that primary surgical decompression improved neurological outcomes in spinal cord compression due to diffuse large B cell lymphoma . In contWith regard to chemotherapy and radiation, the combination of both therapies is more effective than either of them alone. Hyper-CVAD  or CHOP regimens have shown favorable outcomes . The DeAPrimary spinal lymphoma seems to have an unfavorable prognosis. The survival estimates that we found varied. In a series by Flanagan et al, the 2-year survival was found to be 36% . Perry eClassically, central nervous system lymphomas were described in immunocompromised individuals. More cases are being described in immunocompetent patients, thus the need to include this entity in the differential diagnosis of any suspected spinal tumor in all hosts . CliniciWe presented the case of an elderly female who presented with sub-acute back pain and was diagnosed with a primary spinal NHL. The diagnosis of this entity is complicated by the difficulty and delay in obtaining a satisfactory tissue sample. The treatment is incompletely defined due to the rarity of the disease. Physicians should keep this entity in the differential diagnosis of a newly diagnosed spinal tumor in order to hasten diagnosis and potentially improve outcomes.


A 43–years–old woman without any medical history or particular treatment consulted the emergency room for fluctuant headache, developing for five days and resistant to usual treatment. The headache was associated with hypoesthesia of the left part of the face with ptosis. Clinical examination found a slight hypoesthesia in V2 and V3 territories and a Bernard-Horner’s syndrome. Brain magnetic resonance imaging (MRI) showed no brain parenchymal abnormality, especially on diffusion-weighted imaging. Cervical vessels imaging with axial fat suppressed spin echo T1-weighted sequence showed a pre occlusive dissection of petrous segment of the left carotid artery. Study of the cervical portion was suboptimal due to the magnetic susceptibility artifact of a dental implant Figure .A complementary 3D volumetric fat suppressed spin echo T1-weighted sequence was realized and allowed to partially overcome this artifact and to bring out the extension of this dissection to the cervical part of the carotid artery Figure , arrow. Cervical arteries dissections are due to development of hematoma in media layer of the vessel wall. They represent the principal cause of cerebral ischemic strokes in young adults (20%), but the prognosis remains good. They predominate in the carotid arteries, especially in the petrous segment and can be multiple in a third of cases.The role of imaging is fundamental for the diagnosis and the follow-up of both the dissection and the related cerebral ischemic lesions. Indeed, MRI allows at a time analysis of the wall and of the lumen.The axial fat suppressed SE T1-weighted sequence Figure  represenThe authors declare that they have no competing interests.


A 61-year-old woman noticed a right neck lump in October 2001. Fine needle aspiration showed follicular neoplasm, adenoma versus carcinoma. The ultrasound scan showed a solid mass of maximum dimension of 3.7 cm. She had a right thyroid lobectomy and isthmectomy in January 2002 (first surgery). The tissue specimen showed a 4.5 cm Hurthle cell carcinoma (HCC) with vascular invasion. There were no capsular invasion, extra-thyroidal extension, or margin involvement. A completion left lobectomy (second surgery) was performed two weeks later. Therefore the pathological stage is II (T3N0M0). She received adjuvant radioactive iodine ablation for residual thyroid tissue. By 2003, she developed local recurrence, which was resected (third surgery), followed by adjuvant external beam radiotherapy. Unfortunately, she developed further recurrence in the left main bronchus, as identified by Indium-111 Octreotide  and positron emission tomography-computed tomography PET-CT imaging in 2006. She underwent a left pneumonectomy (fourth surgery) in July 2006. In November 2007 she was found to have mediastinal recurrence which was treated with high-dose external beam radiotherapy. She initially responded but developed more local recurrence and a lung metastasis by 2011. She was treated with brivanib with ixabepilone, under a phase I clinical trial with mixed response. Her treatment was discontinued secondary to toxicity and she succumbed to her disease in 2012. This case report illustrates the natural history and clinical decision making for patients diagnosed with HCC of the thyroid. Specifically, we highlight the clinical issues surrounding the histopathological diagnosis, extent of surgical resection, radioiodine diagnostic imaging/ablative treatment, as well as external beam radiotherapy. Hurthle cell carcinoma (HCC) of the thyroid, also called oncocytic carcinoma, is a rare form of differentiated thyroid cancer. Preoperative clinical, cytological, and genetic studies have not been shown to reliably discriminate between benign and malignant variants of Hurthle cell neoplasms (HCN) . TherefoA previously healthy 61-year-old woman noticed a right neck lump in October 2001. The ultrasound scan demonstrated a solitary 3.7 x 3.4 x 3.0 cm heterogeneous solid mass. The family doctor performed a fine needle aspiration in the office that showed follicular neoplasm, adenoma versus carcinoma. The depicted risk of malignancy by Bethesda system is 15-30% [She had a right thyroid lobectomy and isthmectomy in January 2002 (first surgery) because the frozen section could not tell if the tumor was benign or malignant. The tissue specimen showed a 4.5 cm Hurthle cell carcinoma (HCC) with vascular invasion. There were no capsular invasion, extra-thyroidal extension, or resection margin involvement. A completion left lobectomy (second surgery) was performed two weeks later. The pathological stage was II: T3N0M0 due to the maximum tumor dimension of 4.5 cm (TNM 1997 Staging). The histological type was HCC and was confirmed with another pathologist in a tertiary center.As there was a significant amount of residual thyroid tissue by a radioactive Iodine-123 scan, ablation with 100 milliCurie of Iodine-131 was given in April 2002. Unfortunately by January 2003, a doctor felt a 0.6 cm lump in the right neck scar. There was no nodal or distant recurrence with a non-contrast computerized tomography (CT) scan. The Iodine-123 scan did not show any uptake of the radio-isotope in the neck or the rest of the body. The local recurrence in the right neck was locally excised in March 2003 (third surgery) followed by adjuvant radiation 58 Gray (Gy) in 29 treatments in June 2003. A follow-up radioactive iodine scan reported no uptake in the neck including the scan in November 2005. However, serum thyroglobulin gradually rose with time: November, 2003: 4.0; June, 2005: 76.8; November, 2005: 642.2 pmol/L . Serum thyroglobulin antibodies were not present. An Indium-111 Octreotide scan  in January 2006 showed uptake at the level of the bifurcation of the left main bronchus. While on thyroxine, she underwent a positron emission tomography/ computerized tomography (PET/CT) scan in March 2006, which showed a 2.2 x 2.7 cm left hilar mass and a small 8 mm pre-carinal node. The lung fields were clear. No other abnormalities were found. She saw our thoracic surgeon who felt that a left pneumonectomy was required to remove the left hilar mass, which grew to 3.3 x 2.6 cm, and a new 1.2 x 0.6 cm left lower lobe metastasis seen in the June 2006 CT scan. She then underwent left pneumonectomy in July 2006 (fourth surgery). A pathology slide of the lung specimen is shown in Figure By November 2007, a CT scan detected a 2 cm left mediastinal recurrence, but the patient initially declined treatment due to fear of complications. Finally, she agreed to treatment by palliative radiotherapy 60 Gy in 30 fractions to the left upper chest for the mediastinal recurrence, which was finished in October 2008. There was no overlap with the previous radiotherapy fields. The left mediastinal soft tissue recurrence around the aortic arch initially responded in follow-up CT scans, but began to regrow by December 2011 to 3.5 x 3.7 cm  with a new chemotherapy (ixabepilone). She was admitted due to bladder infection and febrile neutropenia. A CT scan in May 2012 showed that the right middle lobe lesion had been reduced to 1.1 x 0.7 cm. The mediastinal mass was stable in appearance. She declined further treatment due to treatment toxicities.By November 2012, the CT scan showed that the right middle lobe mass had grown to 1.9 x 2.4 cm and the mediastinal mass around the aortopulmonary window to 7.3 x 5 cm. There were also a new 3 x 2.5 cm right peribronchial mass and a 4 x 4.9 cm infra-carinal mass. She died of progressive disease by early December 2012.The case report illustrates the decision-making process in HCC. HCC is a less common type of differentiated thyroid cancer. It has distinct adverse features: higher rate of regional lymph node metastases, recurrences and cause-specific mortality, all shown in this case . Four maThe first issue is about the difficulty to separate HCC from other benign or malignant thyroid neoplasms preoperatively or even by frozen section like our case. Hurthle cells (HC), or oncocytes, are large cells found in the thyroid gland and elsewhere, and are characterized by abundant, granular, eosinophilic cytoplasm which is a consequence of the accumulation of altered mitochondria. Pathological differential diagnoses include medullary carcinoma and papillary thyroid carcinoma. Workup typically involves a fine needle aspiration biopsy (FNAB). The Bethesda system for reporting thyroid cytopathology classifies HCC in the diagnostic category of “follicular neoplasm or suspicious for a follicular neoplasm” . HistoloThe second issue is about the extent of surgical resection. Our case had completion thyroidectomy very soon after the pathology report came back as HCC. An ipsilateral neck lymphadenectomy is recommended for clinically/ radiologically/ biopsy-proven lymphadenopathy. Like our case, all patients should have a radioiodine scan three to four months after surgery. The iodine-123 scan was a diagnostic scan to see any residual thyroid tissue. Since there was a lot of residual thyroid tissue, she had I-131 ablation with 100 mCi, followed by a whole body scan nine days later.The third management issue is on imaging and the role of radio-iodine diagnostic scan and treatment. The 2016 National Comprehensive Cancer Network (NCCN) guidelines recommended consideration of radio-iodine therapy based on pathological findings and postoperative serum thyroglobulin levels . Except In patients with a negative radioiodine scan and rising serum thyroglobulin, PET/CT can be ordered. A meta-analysis of seven trials showed an improved detection rate of recurrence/metastasis by either thyroxine withdrawal  or recomThe last issue is on the role of external beam radiotherapy. Our center employed this for gross postoperative residual disease and in this case for adjuvant and salvage treatments. In the modern era, our center also arranges radiofrequency ablation or stereotactic body radiotherapy for limited small lung metastases. Other salvage treatments include ethanol ablation, radioiodine ablation, external radiotherapy, radioisotope and systemic treatment. Our patient had external radiotherapy and systemic treatment.The reported case illustrates the management of this rare cancer. The patient presented with a neck mass, which was investigated with an ultrasound scan. We tried to establish the pathological diagnosis by a fine needle aspiration biopsy and a frozen section of the resected tissue, both of which could not confirm malignancy. The detailed examination found vascular invasion and concluded with the diagnosis of HCC. We performed radioactive iodine ablation, multiple surgeries, external radiotherapy and finally enrolled her into a clinical trial of systemic therapies due to lack of options. Despite our impeccable follow-ups, assessment, and treatments, she suffered multiple recurrences in different locations. Sadly, we lost her after 11 years of the cancer battle. The discussed strategic diagnosis and treatment plans could be used for other cases with successful outcomes. Thus, these approaches will provide insights to the medical and clinical science community.


Few studies have used fine-needle aspiration cytology for the purpose of isolated skeletal Rosai–Dorfman diseases (RDDs) diagnosis.Herein, we described an extremely rare case of a 56-year-old woman who presented to our hospital with an insidious onset of pain in the right proximal tibia.The provisional cytologic diagnosis of RDDs was confirmed by a computer tomography-guided core needle biopsy of the lesion. Subsequently, curettage and autogenous iliac crest bone graft were performed successfully. At the 4th year of follow-up her clinical symptoms disappeared, and there was no clinical evidence of lesion recurrence.Our case highlighted the role of fine-needle aspiration cytology with immunohistochemical studies in the diagnosis of RDDs and the characteristic features of isolated skeletal RDDs in an unusual location, the knowledge of which would help avoid missed or delayed diagnosis in the future. These difficulties were accompanied by progressive pain in the right proximal tibia which treatment with a nonsteroidal antiinflammatory drug did not improve. She denied trauma, fever, chills, skin rashes, night sweats, and weight loss. Physical examination at the time of admission was remarkable only for an isolated, firm, tender, nonfluctuating, nonerythematous, soft-tissue swelling directly over the right proximal tibia without a noticeable deformity, and peripheral lymphadenopathy. Laboratory examination revealed a moderately elevated erythrocyte sedimentation rate 27 mm/h). Other values, including white blood cell count, C-reactive protein, rheumatoid factor, tumor markers , and urinary routine test were within normal ranges. Plain radiograph of the right knee showed a lytic, destructive lesion with sharp border in the proximal tibia  on the cytologic material revealed that numerous large histiocytes had been interspersed with different amounts of lymphocytes, neutrophils, and plasma cells. The cytologic features were suggestive of RDD. Subsequently, the biopsy sample was stained with H&E, and immunohistochemical studies were performed. The H&E of the concurrent core needle biopsy revealed the classic features of RDD Fig. ; similarCurettage and autogenous iliac crest bone graft, without radiation therapy and chemotherapy, were advised to perform, since progressive pain in the right proximal tibial lesion and concern for a potential future pathological fracture. Radiographically at the end of 6 months significant reduction in the size of the lesion was noticed and complete resolution of plain X-ray abnormalities when first seen was observed at the end of 1 year, with no evidence of lesion recurrence during 4 years postoperative follow-up.1 Currently, the origin of this condition is unclear; however, it is suspected to be caused by a virus.4 As per the World Health Organization classification of tumors, these lesions are categorized as a reactive condition of unknown etiology.5 Extranodal RDD accompanied by lymphadenopathy is seen in almost half of the cases; however, extranodal manifestation of RDD in the absence of lymphadenopathy is extremely rare.6 The skin, respiratory tract, orbital cavity, and the central nervous system are the main extranodal sites involved, followed by the skeleton.2 The occurrence of RDD along with isolated skeletal lesions is remarkably rare and is seen in ∼2% of cases, and solitary bone involvement in the absence of lymphadenopathy has been noted in extremely few cases.7RDD (sinus histiocytosis with massive lmphadenopathy) was first described in 1969 by Rosai and Dorfman as benign, idiopathic, distinct histiocytic disorder that is comparatively rare and is typified by painless bilateral cervical lymphadenopathy together with pyrexia, leukocytosis, and increase in erythrocyte sedimentation rate.3 To the best of our knowledge, many studies have used FNAC for the purpose of extranodal RDD diagnosis. Although many clinical disorders and diseases of the skeletal system have been described, only 14 cases of isolated skeletal lesions in the absence of lymphadenopathyh have been reported until date; of these, all except 3 by FNAC were diagnosed on the basis of histologic material.3 An additional case of extranodal RDD as solitary lesion of the bone is discussed here; we feel that this case is the earliest reported case of RDD that occurred in the proximal tibia and was diagnosed using FNAC.The differential diagnosis of extranodal RDD of the bone is occasionally difficult because of the occurrence of clinical signs and symptoms that are nonspecific and because of lesion rarity and the less classic radiologic features observed at times.8 Characteristically, those histiocytes contained abundant cytoplasm and distinct nucleoli with fine chromatin, and demonstrated conspicuous emperipolesis of lymphocytes, plasma cells, and nuetrophils.9 Immunohistochemically, the large histiocytes were intensely positive for S-100 protein, weakly positive for CD68 protein, and remained negative for CD1a protein, which confirmed RDD diagnosis.In the literature that reported the cytologic features of the osseous lesions, classic features of RDD were well described, which was also seen in our case where numerous large histiocytes had been interspersed with different amounts of lymphocytes, neutrophils, and plasma cells.9 A variety of ways have been used and described for the treatment of RDD involvement of the bone, including corticosteroids, chemotherapy, radiotherapy, surgical curettage, or resection; recommended therapeutic modality, if possible, contains the clinical observation.10 As in our case, in the rare case of extranodal site presenting with progressive pain and the difficulty in walking, surgical curettage and autogenous iliac crest bone graft may be necessary. Importantly, surgical intervention seems to be the preferable treatment option for symptomatic RDD, as most cases of isolated skeletal RDD in previously reported studies healed completely after the totally surgical curettage or resection.10 Simply put, skeletal involvement of RDD is seldom fatal, and the long-term prognosis is good for isolated extranodal lesion.6Although the lesion usually undergoes spontaneous resolution, most cases of RDD typically follow a waxing and waning of the clinical course.To our knowledge, there have been no reports describing isolated skeletal RDD that occurred in the proximal tibia and was diagnosed using FNAC. We demonstrate the characteristic features of isolated skeletal RDD in an unusual location and recommend that FNAC with immunohistochemical studies can be a good option in the diagnosis of extranodal RDD of the bone, the knowledge of which would help avoid delayed or missed diagnosis in the future.


Pituitary apoplexy is a clinical syndrome characterized by acute headache, visual changes, and decreased consciousness occurring in association with hemorrhage or infarct of an existing pituitary adenoma. Surgical management involves tumor resection and decompression of surrounding structures including the optic apparatus. Vasospasm is a rare but potentially devastating complication of pituitary apoplexy. We present a case of pituitary apoplexy in a 28-year-old male treated with emergent endoscopic transsphenoidal resection. On postoperative day seven, following  surgical resection, the patient developed neurologic deficits and motor weakness, and severe vasospasm was diagnosed. This is a novel case of intra-arterial verapamil and angioplasty used to treat vasospasm following surgical decompression for pituitary apoplexy. The patient experienced complete recovery of motor deficits following treatment. The authors propose angioplasty as an effective treatment for postoperative vasospasm following transphenoidal surgery for pituitary apoplexy in the presence of focal vessel stenosis. Pituitary apoplexy is a clinical syndrome associated with hemorrhage or infarct of an existing pituitary adenoma characterized by some combination of acute severe headache, visual impairment, ophthalmoplegia, pituitary hormonal dysfunction, and altered mental status -2. ManagHistory and examinationThe patient, a 28-year-old male with a history of migraines, presented to an emergency department complaining of acute visual change, frontal headache, nausea, and vomiting with worsening symptoms over two weeks duration. On physical examination, the patient was completely blind with no light perception in both eyes; his pupils were 4 mm and sluggishly reactive to light, and a left cranial nerve (CN) VI palsy was noted. The patient was otherwise neurologically intact. Magnetic resonance imaging (MRI) demonstrated a heterogeneously enhancing 4 x 3.2 x 2.2 cm sellar mass with 4 cm extension into the adjacent suprasellar compartment. The mass demonstrated compression and elevation of the optic chiasm, and extensive leptomeningeal enhancement. The patient was subsequently transferred to our institution. At the time of our evaluation, the patient continued to have poor vision with persistent absence of bilateral light perception and a left CN VI palsy. Computed tomography (CT) at our institution showed a large sellar and suprasellar mass measuring 4 cm x 4 cm, displacing the third ventricle, extending into the left cavernous sinus, and eroding into the superior aspect of the clivus Figure . LaboratOperative courseGiven the patient’s acute bilateral vision loss, urgent endoscopic endonasal resection of the pituitary tumor was performed using standard image-guidance techniques. A cerebrospinal fluid (CSF) leak was noted postoperatively, which was repaired with Gelfoam, Durepair, and Surgicel. Histologic analysis revealed a pituitary adenoma with necrosis and hemorrhage Figure . PostopePostoperative courseThe patient’s postoperative course was marked by slow improvements in vision and near complete recovery of left-sided motor strength. At the five-month follow-up, continued improvement in vision was noted and he was free of any other neurological deficits. MRI showed minimal residual disease versus postoperative scarring in the sella Figure . An ophtPituitary apoplexy followed by cerebral infarction is a rare occurrence. The mechanism of infarction may be attributed to vasospasm, mechanical compression of an intracranial vessel, or hypothalamic injury .Vasospasm may be the result of subarachnoid hemorrhage or release of vasoactive substances from the tumor . The vasVasospasm has also been reported as a consequence of pituitary tumor resection. Due to tumor proximity to the Circle of Willis and internal carotid arteries, vasospasm may be induced by surgical manipulation , 6. In 2The treatment options for vasospasm following pituitary surgery include hemodynamic therapy, intra-arterial vasodilators, or angioplasty , 7. MediWe report a novel case of vasospasm that developed following transsphenoidal decompressive surgery for pituitary apoplexy. Our patient was treated with intra-arterial verapamil and angioplasty and experienced complete recovery of motor deficits. The authors emphasize the importance of recognizing that vasospasm can be a serious, delayed complication of apoplexy or pituitary surgery. This diagnosis should be considered and treated aggressively and with urgency once diagnosed. We propose a novel treatment for postoperative vasospasm following transphenoidal surgery for pituitary apoplexy, which was extremely successful in this reported case. The success achieved in the cases was made possible by the collaborative efforts of tumor surgeons, endovascular surgeons, critical care physicians, and endocrinologists.


Closed gastroschisis is a rare entity usually associated with intestinal atresia and short bowel syndrome. We report two cases of closed gastroschisis presenting with neonatal intestinal obstruction and para-umbilical evisceration without an abdominal defect. Gastroschisis is a congenital abdominal wall defect that is usually located on the right side of the umbilical cord, rarely; it is located in a mirror image position on its left side. Intestine, stomach, and occasionally other abdominal organs e.g. ovaries eviscerate through that defect [1]. In very rare cases, the defect itself closes around the eviscerated organs causing exit and/or entry level ischemia; a state which has been termed closed or closing gastroschisis and is representing around 6% of all cases of gastroschisis [2]. Sometimes the whole midgut is lost, a condition termed vanishing midgut [3].Case 1:A full term 3 kg male presented with extra-abdominal bowel remnant just to the left side of a normally appearing umbilicus . Antenatal scan prior to delivery revealed intra-abdominal dilated bowel loops. Clinically, there was upper abdominal distension and bilious vomiting. Initial resuscitation was initiated. Abdominal X-ray showed suspicion of jejunal obstruction. Laboratory investigations were within normal limits. Echocardiography showed small patent foramen ovale. The baby was explored on day 2 via circum-umbilical incision. There was small bowel atresia. Remaining small bowel was 30 cm of blind-ended dilated proximal jejunum and 40 cm of small caliber distal ilium. Two fibrous strings were connecting the mesentery of both jejunum and ilium to an area in the inner aspect of the abdominal wall corresponding to the extra-abdominal bowel remnant. There was no abdominal wall defect. There was associated colonic atresia type IIIa at the level of transverse colon. The extra-abdominal bowel remnant and the connecting fibrous strings were excised. Short tapering of the proximal dilated jejunum, then primary end to end anastomosis was fashioned. Double barrel colostomy was done at the site of the colonic atresia leaving a length of 70 cm viable small bowel in addition to an intact ileocecal valve. Oral feeding was started on 10th postoperative day which was increased gradually to full feed on day 28. He was discharged on day 30.Case 2:A preterm (33weeks) male neonate weighing 1.9kg presented with extra-abdominal bowel remnant, 6 cm long, located to the right margin of an intact umbilical ring. Antenatal scan at 30 weeks of gestation showed dilated bowel loops. Follow-up scan performed 2 weeks later showed progressive dilatation of bowel loops without any abdominal wall defect. The baby developed neonatal intestinal obstruction. X-ray abdomen was suggestive of jejunal obstruction. On exploration, the jejunum was ending blindly 20 cm after the DJ junction. The eviscerated bowel remnant was connected from inside with the colon by a short fibrous stalk. There was 30 cm of non-atretic colon until the peritoneal reflection. Primary end-to end jejuno-colic anastomosis was performed. The patient was put on management for short bowel. One month later, the patient was re-explored due to partial intestinal obstruction. Second exploration revealed an ectatic poorly functioning jejunal loop. Side to side jejunocolic anastomosis was done for better bowel drainage. The baby died at the age of 4 months due to cholestatic liver failure and sepsis a complication of TPN.Spontaneous prenatal closure of the anterior abdominal wall defect in gastroschisis is very rare. Closure of the defect can be complete or incomplete with variable sequelae affecting bowel loops [4]. In cases of mild affection, there is a closing abdominal defect with viable bowel. Next in severity are cases with complete closure of the defect with a tiny stalk connecting the intra-abdominal bowel to an extracorporeal bowel remnant. These forms are termed closing gastroschisis and closed gastroschisis [2].The sequence of events is not well known. It is not clear whether the closure of the defect is the primary event or occurs secondary to loss of the eviscerated bowel. Bhatia and colleagues in1996 proposed 3 scenarios [5]. All proposals attributed bowel atresia to vascular insult. First, abdominal wall defect seen early during pregnancy may close spontaneously around the herniated bowel and the supplying superior mesenteric artery resulting in strangulation and necrosis of the midgut. Second, the herniated fetal bowel may incarcerate within the defect leading to its ischemia and atrophy with subsequent closure of the defect. Third scenario starts by volvulus of the midgut with subsequent infarction, resorption, and closure of the defect.Houben et al in 2009 clarified the criteria for successful antenatal ultrasonographic follow up in patients with gastroschisis [6]. They stated that after diagnosis of gastroschisis, fetal progress is monitored at 4 to 6 weeks intervals. More frequent scans were required in cases of intraabdominal bowel dilatation (>10 mm) or growth restriction of the fetus. The key for diagnosing closing gastroschisis is increased or persisted intraabdominal bowel dilatation with hyperperistalsis. At the same time, there is shrinkage or no increase in the size of extracorporeal bowel. After birth, they defined closing gastroschisis as circumferential or partial (>50%) closure of the ring around the protruding bowel associated with intestinal atresia, bowel ischemia, bowel necrosis, or viable intestine. No defect could be detected in our cases that could be explained by the delay in the initial ultrasound; however, progressive intraabdominal bowel dilatation was detected.Infants suffering gastroschisis and vanishing midgut have nearly 70% mortality. Recently, marked improvement in the outcome has occurred due to advances in neonatal care and nutritional support and the introduction of home TPN regimens for pediatric age group [7]. Our survived case had adequate bowel length; 30 cm jejunum, 40 cm ilium, in addition to the viable part of the colon and ileocaecal valve. However, our second case succumbed to complications of management of short bowel syndrome.Source of Support: NoneConflict of Interest: None


Gossypiboma or textiloma is the result of a foreign-body reaction to extraneous material, usually a surgical sponge that was accidentally retained within the body.The diagnosis of a retained surgical sponge is often delayed due to its infrequent occurrence and protean appearances.The purpose of this pictorial review is to define the common sonographic and CT features of gossypiboma. A retrospective review of sonographic and CT images of 6 surgically proven cases of retained surgical sponges was undertaken. Gossypiboma or textiloma is the result of a foreign-body reaction to extraneous material, usually surgical sponge that was accidentally retained within the body. This is most often associated with emergency procedures, prolonged surgeries, unexpected changes in the course of operation, excessive haemorrhage, change of personnel during the operation or inexperienced and inadequate number of staff .Radiologically, foreign bodies can imitate tumours or abscesses. The manifestations of gossypibomas are protean, and diagnosis is often difficult. Gossypibomas are associated with severe complications leading to high patient morbidity. Moreover, gossypibomas have significant medicolegal implications .Gossypibomas are frequently diagnosed in the intraabdominal cavity. However, they can also be found in the chest ,4, extrePathologically, gossypibomas can produce two types of reactions in the body. One is an aseptic fibrinous response that results in adhesions or encapsulation leading to granuloma formation. The other is of an exudative nature leading to abscess formation, which may be complicated by secondary bacterial infection and fistula formation. The latter usually presents during the early post-operative period.Radiological characteristics of gossypibomas are variable.Sonography: Ultrasound often demonstrates a well-delineated mass containing a whorl-like, hyperreflective echo with a hypoechoic rim and strong posterior acoustic shadowing. Ultrasound characteristics can be classified into two types, a cystic type and a solid type. The former presents as a cystic lesion with a wavy echogenic structure within. The solid type can appear as a complex mass containing hyper- and hypoechoic regions. The retained material itself usually causes acoustic shadowing on ultrasound, although it may be due to calcified areas in the gossypiboma or pockets of air.CT scan: CT may show a low-density, heterogeneous mass with a thick peripheral rim. The presence of mottled, bubbly gas shadows should prompt the diagnosis. The spongiform pattern with gas bubbles  is oftenGossypibomas may give rise to various complications depending on the type of pathologic response. While fibrinous reaction results in encapsulation and localisation, an exudative reaction will lead to extrusion of the foreign material through a fistulous track, either externally to the skin or internally into the rectum, vagina, bladder, or intestinal lumen . This caAbdominal gossypiboma following surgery for ruptured ectopic pregnancy. A 31-year-old female underwent surgery for ruptured ectopic pregnancy. One week later, she presented with vomiting and abdominal pain .A 55-year-old woman with retroperitoneal gossypiboma .A 45-year-old male with low-grade pain in the upper abdomen following gastrojejunostomy .Intrauterine gossypiboma in a 23-year-old woman with pelvic pain and suppurative discharge 3 months after caesarean section .Pelvic gossypiboma complicated by a fistulous connection with the caecum. A 40-year-old woman with pain in the right iliac fossa. The patient had a recent history of hysterectomy .Intraabdominal gossypiboma in a 50-year-old woman with a history of prior surgery for gallbladder carcinoma who presented with abdominal pain and fever .Gossypiboma is not an uncommon entity and the radiologist should be aware of its common and unusual appearances in order to make a correct diagnosis in the appropriate clinical setting.


Embryonal rhabdomyosarcoma (RMS) is a rare type of sarcoma, primarily seen in the pediatric and adolescent population. Three subtypes of embryonal RMS are described, with the botryoid type being the most common. The incidence of this disease in adult females is 0.4% to 1% with the affected age group being patients in the third to fourth decade of life. It is exceedingly rare in patients above 40 years of age. We describe the case of a 48-year-old female, gravida 9 para 5, who presented with abnormal vaginal bleeding and an exophytic mass on examination. Given her lack of requirement of maintaining parity, she underwent radical surgery. The tumor was 8 cm in the largest dimension with a high histologic grade and some cartilaginous differentiation. Immunohistochemical stains were positive for vimentin, CD99, myogenin, and MyoD1 consistent with a diagnosis of embryonal rhabdomyosarcoma, botryoid subtype. Based on high survival rates when treated with aggressive adjuvant chemotherapy, a decision was made to treat the patient with the ARST0331 regimen. We discuss the diagnostic pathologic features of the disease, the epidemiology, and the most common presentation along with prognostic factors, treatment strategies, and outcomes. Rhabdomyosarcoma (RMS) of the cervix is a rare disease entity, particularly in the adult population. The tumor is histologically subdivided into the embryonal, alveolar, sclerosing, and pleomorphic types. While embryonal is the most common histology seen, it also has three subtypes; botryoid, spindle cell, and not-otherwise-specified. Given the rarity of the disease and its histologic variants, standard treatment guidelines are not only scarce but difficult to devise . ReporteA 48-year-old female was referred to the gynecology clinic with symptoms of abnormal vaginal bleeding for the prior three to four months. She was gravida 9 and para 5, with a history of one miscarriage and three elective terminations. Her last visit to a gynecologist was 13 years ago after her last pregnancy. The patient’s past history was significant for hypertension controlled with a calcium channel blocker and she had a laparoscopic ovarian cystectomy several years ago. Her family history was significant for a maternal first-degree cousin with a gynecologic malignancy  diagnosed in her 20s treated with curative surgery. On pelvic examination, she was seen to have blood clots in the vaginal canal. The cervix appeared dilated, with a protruding friable mass that was biopsied with pathology consistent with embryonal rhabdomyosarcoma.The patient was referred to a gynecologic oncologist and further workup revealed the absence of metastatic disease. A decision for surgical intervention was made and the patient underwent a total abdominal hysterectomy, bilateral salpingo-oophorectomy, omentectomy, left pelvic lymph node biopsy, and right ureterolysis. The pathologic exam showed a tumor measuring 8 x 3 x 1 cm with a high histologic grade seen to be invading the cervical stromal connective tissue with areas of cartilaginous differentiation. No lymphovascular invasion was observed and the tumor margins were uninvolved by sarcoma. On immunohistochemistry, the cells were strongly and diffusely positive for vimentin, positive for CD99, and focally positive for myogenin and MyoD1. The findings were consistent for a diagnosis of embryonal rhabdomyosarcoma of the cervix. The pathologic stage was pT1a, i.e., tumor limited to the endocervix without myometrial invasion  and no further recommendations were deemed necessary.Based on the Intergroup Rhabdomyosarcoma Study Group (IRSG) classification , the patRhabdomyosarcomas (RMSs) are a group of tumors that arise from immature cells destined to form striated skeletal muscle. Nearly 90% of all cases of RMSs are seen in individuals less than 25 years of age, and within this age group, 60% to 70% of the patients are less than 10 years of age. Whereas RMS accounts for 3% to 4% of all childhood cancers, it is seen with an incidence of less than 1% in adults [RMS can be subdivided into four types: embryonal, alveolar, sclerosing, and pleomorphic. The embryonal RMS is the most common type, accounting for 68% of all RMSs and is subdivided into three categories: the botryoid, spindle cell, and not-otherwise-specified (NOS). The majority of RMSs arising from the female genital tract is of the botryoid type. The term botryoid is derived from the appearance of the tumor - that of grape-like structures. Since it arises in the cervix, it grows in the narrow space and takes the appearance of a grape-like mass. On histology, the cellular pattern is variable with the presence of mitoses, pleomorphism, a cambium layer, attenuated epithelium, myxoid type stroma, and the presence of atypical rhabdomyoblasts. On immunohistochemistry (IHC), the tumor is usually seen to be positive for vimentin, desmin, actin, myoglobin, MyoD1, and myogenin. IHC is essential for a confirmatory diagnosis .Among adults, most reported cases have been seen to occur in the reproductive age group in the second and third decades of life. However, embryonal RMS has been reported in females older than 50 years of age. The most common presentation of embryonal RMS is the presence of an exophytic mass and vaginal bleeding. Of the reported cases, tumors as large as 6 cm have been seen to occur, with the smallest being about 1.5 cm. Our patient had a mass that was 8 cm, that is, larger than the mean size reported in most series [The prognosis depends on the tumor site, depth of invasion, and lymph node involvement. Tumors arising from the cervix have a better prognosis than similar tumors arising in other sites of the female genital tract. Extrapolated from findings in RMSs of the bladder and vagina, an exophytic growth pattern is associated with a more favorable prognosis than an endophytic or diffuse intramural pattern. The embryonal botryoid variant is associated with a much more favorable outcome than the alveolar and undifferentiated subtypes, which are associated with a particularly poor prognosis. An early disease stage at diagnosis is another favorable prognostic factor, whereas metastatic disease at presentation and poor response to chemotherapy are strongly associated with a poor prognosis. Adjuvant chemotherapy is decided based on a risk group of low, intermediate, or high. The regimen usually comprises of vincristine, dactinomycin, and cyclophosphamide with modifications in dosing schedule based on the prognostic group . Since cervical sarcomas are a rare entity and histologically diverse, standardized management guidelines for the disease are lacking. Germline DICER1 pathogenic variants have been described in patients with embryonal RMS of the uterine cervix in older children and young adults, which can present as vaginal spotting or passage of myxoid, hemorrhagic solid tissue. It may be prudent to test for the mutation if suspicion of a genetic predisposition exists .Even though embryonal rhabdomyosarcoma is an uncommon disease of the pediatric and adolescent population, the disease must be kept in the differential diagnosis of an older female presenting with vaginal bleeding or a protruding mass. Accurate diagnosis can be established by performing appropriate immunohistochemical stains. In addition, treatment strategies for a rare disease, such as cervical RMS, are devised from experience with comparable cases having analogous tumor pathology. Reporting of the presentation and outcomes of these cases is essential in making informed therapeutic decisions for patients with RMS of the cervix.


Complete hindgut duplication is a rare and intriguing entity, often coupled with genitourinary abnormalities and neural tube defects. The diagnosis demands a thorough clinical exam and radiological workup. Timely recognition and expeditious treatment of these patients can lead to a better quality of life. We present a case of a 10-month-old female with complete hindgut duplication and associated genitourinary duplication treated with surgical intervention. Gastrointestinal (GI) duplications are a rare occurrence and may involve any part of the alimentary tract. The diverse clinical presentations depend on the size, site, and type of duplication. Complete hindgut duplication, an extremely rare condition, often presents with associated genitourinary duplications.We present a case of total duplication of the hindgut. The patient also had a duplication of the genitourinary system. A review of the literature revealed only a handful of patients with the same condition. One case reported by Schatz had a complete duplication of bladder, urethra, vagina, uterus, and anus . The manA 10-month-old female born at term was brought to the outpatient department by her mother with a complaint of fecal discharge from the vagina since day twenty of birth and constipation with mild abdominal distention for the last two months. The mother had noted fecal discharge from the normal anal opening as well as the vagina. Past medical, surgical and birth history were unremarkable. On examination, the patient was lying comfortably and was responsive to her surroundings. Her vital signs were stable. Abdominal examination revealed soft abdomen with mild distention. There was no tenderness or any palpable mass and no signs of ascites. Bowel sounds were audible. Digital rectal examination (DRE) showed normal anal opening. On rectal exam, a soft bulge was felt on the anterior wall of the rectum with an impression of an extrinsic growth. There was a fistulous opening in the vestibule of the vagina from which fecal matter was exuding. Rest of the systemic examination revealed no abnormalities.Routine laboratory investigations were normal. Contrast enema revealed duplication of the anal canal, rectum, sigmoid colon and situs inversus with liver on the left as shown in Figure On the basis of history, physical examination and investigations, a diagnosis of hindgut duplication were made and examination under anesthesia (EUA) and exploratory laparotomy (EL) was planned. During EUA, six perineal openings  were revealed as shown in Figure On contrast studies two urinary bladders and two vaginas were visible. EL revealed duplication of hindgut from anus till cecum/appendix as seen in Figure Distally, the ectopic anus was excised and proximally the duplicated gut was internally drained into the normal rectum. The septum between the two rectums was divided with linear cutter stapling device to create one single sigmoid and rectum. The septum between the two bladders was divided and converted to a single lumen viscera. The patient had an uneventful postoperative period and was discharged on the fifth postoperative day with a follow-up plan of care.The repair of the double urethra was postponed till the age of three years where the urine continence status of the patient will determine further management. The management of double vagina was postponed till puberty after assessing the menstrual and fertility status of the patient.Gastrointestinal (GI) duplications are a very uncommon, but complex set of embryological disorders can involve any part of the alimentary canal from the tongue to the anus. Duplications of any kind share a few common characteristics. They are hollow, lined with gastrointestinal tract epithelium and have a smooth muscle wall.GI duplications were initially believed to be more common in males as compared to females but it was later confirmed by a study that there is an equal male to female ratio . ColonicColonic duplications can be cystic or tubular involving a limited portion of the colon or be extensive. They are usually divided into two types, type I and II. Type I usually has partial involvement of the colon or rectum while type II has a wider spectrum as in addition to colon and rectum, there can also be associated congenital anomalies including duplication of the lower genitourinary tract, double appendices, situs inversus and neural tube defects .The presentation of this condition might vary greatly from patient to patient depending upon the degree of and site of duplication. In a colonic or anal duplication, the patients are usually asymptomatic. If symptomatic, a colonic duplication can cause symptoms related to an obstruction such as abdominal pain, distention, and constipation. There can be concurrent fistulas in tubular colonic duplications with other surrounding structures or hollow viscera such as vagina, vulva, perineum and urinary tract. Without a fistula, there can be a distention of the duplicated gut distally. In our patient, there was a presence of a recto-vaginal fistula, which presented as fecal discharge from the vagina initially. The patient also had mild progressive abdominal distention afterward.Urogenital duplication can present as a separate entity or in association with hindgut duplication. The constellation of these associated abnormalities can be explained by "partial twinning" of the caudal end of the embryo or abnormal development of urorectal septum . DuplicaVarious diagnostic modalities are available for the diagnosis of GI duplications. Ultrasonography and contrast studies are most widely used. Computed tomography (CT) and magnetic resonance imaging (MRI), although less often used, are helpful in localizing and diagnosing complex duplications . TreatmeAlthough extremely rare, hindgut duplications, if found, should be investigated and a clinical-radiological correlation should be made to accurately delineate the site and extent of the disease. Also, one must be vigilant, as these duplications are often associated with other systemic anomalies. Management of the duplication depends upon the extent and symptomatology of the disease, as larger and symptomatic duplications require more aggressive surgical management.