Renal cell carcinoma (RCC) has potential to present with distant metastasis several years after complete resection. The common sites of metastases include the lungs, bones, liver, renal fossa, and brain. RCCs metastasize rarely to the duodenum, and duodenal metastasis presenting with acute gastrointestinal bleed is infrequently reported in literature. We present a case of synchronous presentation of duodenal and azygoesophageal metastasis manifesting as acute upper gastrointestinal bleeding, four years after undergoing nephrectomy for RCC. The patient underwent further workup and was treated with radiation. The synchronous presentation is rare and stresses the importance of searching for recurrence of RCC in patients presenting with acute gastrointestinal bleeding. Malignancy accounts for about 5% of upper gastrointestinal hemorrhage and may need surgical intervention in the majority of cases. Various segments of the upper gastrointestinal tract can be involved, but duodenal involvement is very rare.RCC accounts for approximately 3% of all malignancies in adults leading to approximately 13,000 deaths annually in the United States of America. Renal cell carcinoma has a strong tendency to metastasize many years following surgical resection. Metastatic sites for RCC are the lungs, bone, liver, adrenal glands, and brain; however, gastrointestinal tract can be involved for solitary late recurrence in rare instances. Duodenal metastasis of RCC has rarely been reported in the literature, but synchronous tumor presence in azygoesophageal recess and duodenum presenting as melena was never reported in literature to the best of our knowledge.We present the case of a 74-year-old man who presented to our primary care clinic with an episode of dark colored stool. He denied any other gastrointestinal symptoms at the time of presentation. He reported subjective weight loss in the last few months. He was diagnosed with RCC four years ago when he was noted to have an incidental renal mass on computerized tomography (CT) of the abdomen. A total nephrectomy was performed revealing renal carcinoma with clear cell subtype on histopathology . FuhrmanHe was also known to have hypertension, type II diabetes mellitus, coronary artery disease, asthma, gout, chronic kidney disease stage II, and benign ethnic leukopenia. He was a former smoker with a 20-pack-year history of tobacco smoking. His father was diagnosed with colon cancer at the age of 74.On initial evaluation his vitals were temperature 98.6 F, pulse 88/minute, respiratory rate 15/minute, and blood pressure 130/88 mm Hg. He was obese with BMI of 30 and had conjunctival pallor on general physical examination. There was bilateral air entry on auscultation of lungs with no adventitious sounds. Precordial examination revealed normal heart sounds with no murmur, rub, or gallop. His abdomen was soft without any tenderness with no palpable masses. Rectal examination was significant for decreased sphincter tone and external hemorrhoids. Laboratory evaluation showed iron deficiency anemia, with ferritin level of 11 micrograms/liter. Serum chemistry showed elevated creatinine of 1.9 gm/dL consistent with his chronic kidney disease. His liver chemistries were within normal limits.He underwent emergent upper gastrointestinal endoscopy and colonoscopy. Colonoscopy showed colonic diverticulosis and a few hyperplastic polyps in the rectum and sigmoid colon. Endoscopy showed duodenal nodule that was biopsied and histopathology revealed duodenal mucosa showing nests and clusters of neoplastic cells with clear cytoplasm in the lamina propria, consistent with metaplastic RCC Figures  and 3. APatient underwent workup for staging with CT scan of the abdomen and pelvis showing normal appearing right kidney. A 3.5-centimeter mass was noted in the azygoesophageal recess closely approximating the esophagus . There wMultidisciplinary team including surgery, oncology, and radiation oncology evaluated the case to plan further management. In view of metastatic disease and recurrent unstable angina episodes suggestive of severe coronary artery disease, patient was deemed not a suitable candidate for surgical resection. He was started on radiation therapy and subsequently being followed up in the medical oncology clinic.RCC is known to be associated with late recurrence of metastatic disease. Levy et al. reported that 23.8% of patients with RCC developed metastasis years after radical nephrectomy in T2 N0 M0 patients . This riDuodenal metastasis is an uncommon presentation of RCC . There hOur case is unique with synchronous presentation of a duodenal and azygoesophageal metastasis presenting as melena. Management of metastatic RCC is dependent on the site. The treatment options for solitary duodenal RCC metastasis depend upon the extent and location of the lesion and range from metastasectomy to Whipple procedure. In cases of overt GI bleeding attributed to duodenal metastasis, interventional embolization has been reported to control the bleeding effectively. Isolated solitary metastasis of RCC has better prognosis than widespread metastasis, which is highly resistant to chemotherapy and radiotherapy, and the treatment of metastatic RCC currently remains ineffective. Sunitinib is currently the most widely used oral vascular endothelial growth factor receptor kinase inhibitor for the initial treatment of metastatic clear cell RCC . AlthougIf patients with history of RCC have any gastrointestinal symptoms including occult or overt bleeding, obstructive symptoms, or iron deficiency anemia, the possibility of metastasis to duodenal or gastrointestinal tract involvement should be kept in mind nevertheless with a history of nephrectomy. Such patients should be investigated at the earliest with appropriate radiological studies, upper endoscopy, and colonoscopy. Awareness of this entity and a high index of suspicion on the part of the treating physician and pathologist would help in proper diagnosis and treatment.


A 52-year-old female presented with frequent episodes of falls without loss of consciousness. These episodes lasted for brief period followed by full neurological recovery. Magnetic resonance imaging (MRI) of the brain showed foramen magnum meningioma encasing left vertebral artery. The patient had dramatic improvement after excision of the tumor. Meningioma is the commonest tumor in the region of foramen magnum . It ofteA 52-year-old female, a known case of hypertension, presented with sudden episodes of fall without loss of consciousness for the last 5 months. Each episode used to last for approximately 1 minute followed by full neurological recovery. The frequency of such drop attacks was 1-2 per day. It was associated with giddiness. Neurological examination was normal apart from mild hyperreflexia in all four limbs.Twenty-four-hour electroencephalography (EEG) was done by a neurologist which was normal. Electromyography (EMG) and nerve conduction velocity (NCV) of all four limbs were also normal. Cardiovascular workup which included echocardiograms (ECG) and Holter monitoring were normal. Magnetic resonance imaging (MRI) revealed anterolaterally placed, homogenously enhancing dural based lesion in the foramen magnum encasing the left vertebral artery suggestive of meningioma Figures .Patient underwent surgery by posterior approach. Total excision of the tumor followed by augmentation duraplasty was performed via midline suboccipital craniectomy and removal of C1 arch. Tumor was arising from anterolateral dura of foramen magnum, firm in consistency, vascular, and nonsuckable. It was excised in piecemeal, baring the left vertebral artery and left posterior inferior cerebellar arteries (PICA). Complete excision was done with coagulation of dural base.Patient had a dramatic recovery from her symptoms postoperatively. At follow-up of 18 months after the surgery patient is disease-free Figures .Histopathological examination showed the lesion to be meningothelial meningioma .Fall of sudden onset can be associated with or without loss of consciousness. Falls with loss of consciousness can be due to syncope or nonsyncopal conditions like metabolic disorders including hypoglycaemia: hypoxia, epilepsy, and intoxications. Falls without loss of consciousness can be due to cataplexy, drop attacks, psychogenic “syncope” (somatization disorders), and transient ischaemic attacks .Syncope is defined as a transient, self-limited loss of consciousness, usually leading to fall. The onset of syncope is relatively rapid, and the subsequent recovery is spontaneous, complete, and usually prompt. The underlying mechanism is a transient global cerebral hypoperfusion. The causes of syncope include neurally mediated reflex syncopal syndromes, cardiac arrhythmias, orthostatic hypotension, and cerebrovascular conditions .Drop attacks are sudden falls without loss of consciousness that are not precipitated by a specific stimulus, occur with abrupt onset and without warning, and are followed by a rapid return to baseline. A range of localizations for drop attacks is possible, but most commonly lower brainstem or spinal cord structures are implicated. Drop attacks generally indicate transient impairment of bilateral central nervous system structures involved in maintenance of postural muscle tone and balance . TumarkiIn elderly patients with sudden falls and presumed drop attacks, the absence of a history of loss of consciousness is unreliable. More than two-thirds of such patients are in fact found to have forms of syncope, epilepsy, and cerebrovascular disorders like vertebrobasilar insufficiency. Drop attacks due to vertebrobasilar insufficiency are commonly accompanied by other event-related neurologic manifestations  in addition to the sudden loss of postural tone in the legs . TherefoForamen magnum (FM) is bounded anteriorly by lower third of the clivus and upper edge of the body of C2, laterally by jugular tubercles and upper aspect of C2 laminas, posteriorly by anterior edge of the squamous occipital bone and C2 spinous process.Meningiomas of Foramen magnum represent around 3% of all meningiomas and 1% of all primary brain tumors. These represent 70% of all tumors in that region. The lesion is often large at diagnosis because of their slow-growing rate, long interval since the first symptom, and the wide subarachnoid space at this level . These pForamen magnum meningioma may rarely present with drop attacks and should be considered in the differential diagnosis of the conditions causing drop attacks. Magnetic resonance imaging (MRI) may clinch the organic causes of drop attacks and help in early diagnosis of foramen magnum meningioma. It should be included in the diagnostic workup of patients presenting with drop attacks.


Case Report. A 38-year-old female gave an eight-year history of a slow-growing mass on her lower lip with intermittent change in size. She presented with a submucosal nodule and thin overlying mucosa adjacent to the vermilion border. Surgical excision was carried as the diagnostic and therapeutic approach. Conclusion. Lip SCL is rare, and surgical excision is advocated in order to exclude underlying pathology and minor salivary gland tumours.Spindle cell lipoma (SCL) is a rare distinct variant of lipoma, which presents as a painless, circumscribed, slow-growing, superficial lesion on the lip and can mimic a minor salivary gland tumour. We present a slow growing lower lip lesion and its management.  Lipomas are uncommon in the oral cavity and are reported to occur in only 1–4% of cases, with no gender predilection , with a SCLs are typically seen in the neck, trunk, and shoulder region in elderly males –8 in theSCLs usually present as painless, well defined , circumsThey have excellent prognosis with no recurrence after surgical excision , 15–17.We present a 38-year-old Caucasian female, referred to our oral and maxillofacial unit by her general practitioner, with a history of a slow-growing lesion on her lower lip for approximately eight years. She reported intermittent change in size and bleeding during the winter months.She was otherwise fit and well with no known allergies.Socially history revealed she did not smoke and consumed alcohol minimally.On clinical examination, she presented with a soft tissue mass over the midline of her lower lip, measuring approximately 6 × 6 mm that was a pedunculated, submucosal nodule adjacent to the vermilion border.After discussion with the patient, she opted to have the lesion excised for cosmetic reasons. An incision along relaxed skin tension line of the lower lip was made, and an encapsulated soft tissue mass was removed with a cuff of adipose tissue.Differential diagnosis at the time of clinical examination included, mucocele, liposarcoma, and myxoma .Histological findings revealed evidence of a benign encapsulated tumour, composed of lobules of uniform, mature adipose tissue with admixed spindle cell in a fibrous myxoid stroma. The spindle cells stained positively with CD34 but negatively for the S-100 protein. These features confirmed the lesion was in fact a spindle cell lipoma .We carried out a conservative surgical removal of the lesion under local anaesthetic.The patient was reviewed four months after the procedure with no postoperative complications.SCL first described in 1975 by Enzinger and Harvey  is a disIn the English literature, only 35 cases of oral SCLs have been presented to date  with an To our knowledge, only 2 cases of lip SCL have been reported. Billings et al.  reportedSCL is a rare entity, particularly on the lip, as such areas are devoid of fat cells. The most common oral site for SCL is the tongue (37%), followed by buccal mucosa (31%) and floor of the mouth (15%). Other sites including the hard palate and alveolar ridge have also been reported.Aetiology and pathogenesis are still unclear , 14, 17.The occurrence of multiple lipomas is associated with Cowden's syndrome or multiple hamartoma syndrome . MultiplDiagnosis is based on both the clinical and histological characteristics.Microscopically, SCLs do not differ from normal fat cells; however they do differ metabolically  in that Intraoral lipomas are described into the following microscopic subtypes: classic lipoma make up 80%, and other variants such as fibrolipoma, intramuscular lipoma, minor salivary gland lipoma , angiolipoma, and infiltrative and spindle cell lipoma make up the remaining 20%.Histologically, there are two characteristic features associated with a SCL. First, the lesion is composed of bland mitotically inactive spindle cells arranged in parallel registers between the fat cells, associated with thick rope-like collage bundles . The othAnother feature of note is the mast cells which are a feature in almost all SCLs , 19, alsThe treatment of choice of all histological variants is local surgical excision  with gooThis case highlights the fact that SCL should be considered in the differential diagnosis of oral cavity mesenchymal tumours, as well as its malignant counterpart, liposarcoma. It should be noted that although liposarcoma is a common soft tissue neoplasm, its occurrence in the oral cavity is rare . MalignaOther connective tissue lesions such as granular cell tumour, neurofibroma, and salivary gland lesions (mucocele and mixed tumour) might be included in the differential diagnosis .SCL, a distinct histological variant of lipoma, is a rare entity in the oral cavity. It is a benign neoplasm, usually asymptomatic and typically slow-growing. Aetiological factors are unclear and definitive diagnosis is confirmed by both clinical presentation and microscopic findings. Treatment is by conservative surgical excision to exclude underlying pathology and minor salivary gland tumours.Intraoral SCL is a rare clinical presentation and usually presents as slow-growing, well circumscribed lesion.Aetiology is unclear.Accurate diagnosis depends on correct correlation between histological and clinical features.SCL has unique histological and immunostaining characteristics.Treatment is usually conservative excision with no recurrence and excellent prognosis.


Orbital neoplasms with associated bone erosions and intracranial extension are generally considered suspicious for malignancies. Here, we describe the clinical and radiological findings, as well as the surgical management of two extraordinary cases, in which such bony perforations with subsequent intracranial tumor growth resulted from benign angiomatous orbital neoplasms. Two female patients, 69 years old (case 1) and 51 years old (case 2), had both developed visual symptoms  over several months. Computed tomography revealed an orbital tumor of the anterosuperior orbit with painless swelling of the medial upper eyelid of the right eye in case 1, and a posterior intraconal tumor close to the orbital apex of the left eye in case 2, respectively. In both cases, the tumor was associated with a perforation of the orbital roof connecting the orbit with the anterior cranial fossa. An interdisciplinary ophthalmologic and neurosurgical approach allowed for complete tumor removal, in both patients with no signs for local recurrence during the subsequent follow-up of 15 and 18 months, respectively, as well as for a satisfactory visual rehabilitation. Orbital tumors although relatively rare with an estimated incidence of around 3 cases in one million people comprise a variety of benign and malignant neoplastic entities , 2. GiveIn order to choose an adequate diagnostic as well as therapeutic approach, not only tumor dimensions and localization have to be considered but also and as importantly the benign versus malignant character of the lesion. While benign tumors may be observed and assessed conservatively as long as the (bin)ocular function remains unaffected , malignaIn turn, incisional and excisional orbital surgery is demanding, often requiring a multidisciplinary approach of orbitoplastic, neurosurgical, and maxillofacial surgical specialists, and is associated by itself with a significant risk for the ocular function , 8. The Commonly, tumor derived perforations of the bony orbit are among those radiological features raising suspicion for a malignancy, thus also impacting therapeutical decisions. However, very rarely bony erosions and intracranial tumor extension may derive from benign orbital tumors as well, which we demonstrate with this series of two patients, who presented between 09/2013 and 03/2014 to our oculoplastic and ophthalmo-oncological service at the Department of Ophthalmology, University of Cologne, Cologne, Germany, with anterior (case 1) and posterior (case 2) orbital neoplasms of primarily uncertain behavior.A 69-year-old female patient was referred to our hospital in 09/2013 with a tumor of the anterosuperior orbit accompanied by a pea-sized livid painless swelling of the medial upper eyelid of the right eye. Based on the history and portrait photographs, a significant increase in tumor size could be assessed over an 18-month-period. According to the anterior localization in the orbital entrance Hertel exophthalmometry revealed no protrusion of the ipsilateral eye globe. Functionally, the tumor did not affect best-corrected visual acuity  or ocular motility. However, swelling of the upper eyelid (Fig. A 51-year-old female patient presented to our clinic in 03/2014 reporting a progressive visual impairment in her left eye over 6 months. BCVA was reduced to 20/40 and 30° perimetry showed a concentric visual field restriction. Hertel exophthalmometry showed a slight 2 mm proptosis of the left eye (Fig. Vascular tumors, particularly cavernous hemangiomas in adults and capillary hemangiomas in infants, belong to the most frequent primary benign orbital neoplasms . As the Among the mechanisms, which might contribute to bone destruction adjacent to benign orbital tumors, a pressure-induced atrophy might as well come into question as a pro-osteoclastic growth factor milieu derived from tumor surrounding inflammatory cells .Bone erosions related to tumoral orbital lesions, even though remaining suspect for malignant neoplastic entities may be the result of angiomatous benign lesions. Our cases and the few previously published ones underline that the assumed diagnosis, the differentiation between benign and malignant entities, and the subsequent urgency for surgical interventions should be based on as many clinical and radiological criteria as available, rather than on isolated findings that might lack sufficient sensitivity and specificity .If surgery of orbital neoplasms with bone perforations and intracranial extension is intended, a more complicated and prolonged surgical course should be anticipated, since stronger tissue adhesions might be found. Since little invasive extracranial approaches usually performed by orbital and ophthalmic plastic surgeons are unsuitable to make such tumors accessible and resectable as a whole, transcranial interdisciplinary approaches are mandatory, allowing for complete tumor removal as well as for rehabilitation of visual functions.Written informed consent was obtained from the patients for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.


To report a rare case of a recurrence of spiradenoma that developed in the upper eyelid.A 49-year-old woman who had a second recurrence of a tumor in the right palpebral conjunctiva underwent local resection of the lesion with adjunctive cryotherapy to the surgical site. The tumor consisted of smooth, round to oval nodular lesions approximately 1–3 mm in size with enlarged blood vessels. Histopathologically, the solid and well-circumscribed nodule was located beneath the conjunctival epithelium. It was composed of cells with slightly basophilic-to-clear cytoplasm and round-to-oval nuclei arranged in a trabecular pattern. Periodic acid-Schiff stain was positive in the cytoplasm, and the staining disappeared after digesting by diastase. Many cells in mitosis were observed throughout the tumor but no necrotic cells. Immunohistochemistry showed that the Ki-67 labeling index was 12%. From these findings, we diagnosed this tumor as a recurrence of the spiradenoma. There has been no recurrence and no signs of malignancy in the 6 months after the surgical excision.Our findings indicate that a spiradenoma should be completely excised surgically because of malignant transformation after repeated recurrences. Tumors arising from the sweat glands of the eyelids are uncommon, and the differences in their structure have given rise to much confusion -4. A spiA 49-year-old woman visited a neighborhood eye clinic complaining of a foreign body sensation and pain in her right upper eyelid of several days duration. She was referred to our hospital for further evaluation and treatment on the next day. Several years earlier, she had a tumor excised from her right eyelid, and the tumor was diagnosed as a spiradenoma. Our examination showed that her best-corrected visual acuity (BCVA) was 20/30 OD and 20/20 OS. A round to oval-shaped nodule with smooth borders was present in the right upper palpebral conjunctiva Figure A, which A spiradenoma is a tumor of the eccrine sweat glands that arises as a solitary, intradermal, and painful nodule on the chest or face and most frequently in early adulthood. Occasionally, there may be multiple tumors ,2,5. It Immunohistochemical study demonstrated that cytokeratin expression in eccrine spiradenoma was compared with expression in normal eccrine glands and the tubular structures are CEA positive ,7. In thThe differential diagnosis includes cylindroma, hidradenoma and sebaceoma. Dermal cylindroma and spiradenoma in the same patient or in the same tumor mass are well-represented in the literature ,8. CylinMalignant changing of spiradenomas is unusual, and the histology of malignant spiradenomas resembles squamous metaplasia or very poorly differentiated pseudosarcomatous spindle cell elements . In caseWe describe a recurrence of spiradenoma due to an inadequate surgical excision at a rare site, the palpebral conjunctiva. Although a spiradenoma is usually benign, it may transform to a malignant spiradenoma, and thus we are following this patient carefully.The study was approved by the Ethics Committees of Gifu University Hospital. Written informed consent was obtained from the patient for publication of this Case report and any accompanying images. A copy of the written consent is available for review by the Series Editor of this journal.The authors have no proprietary or financial interest in any products used in this study.Conceiving and designing the study (MK); data collection ; interpreting the data ; writing the manuscript (SA), providing critical revisions  and approving the final version . All authors read and approved the final manuscript.The pre-publication history for this paper can be accessed here:http://www.biomedcentral.com/1471-2415/14/85/prepub


This 32-year-old-man was admitted to the emergency department because of S1 left acute sciatica. An emergency CT-scan showed a highly probable appearance of herniated disc in L5-S1 level (A). The patient was operated without finding disc herniation, but an aspect of anterior and posterior epidural abscess compressing the dural sheath at L5-S1. Medullary MRI showed after the up and the down extention of disease process (B). Histological examination of samples found a B lymphoblastic leukemia (C). The patient died 3 weeks after because of systemic infection. After review of literature, it was never been described that the sciatic pain can be a mode of revelation of B acute lymphoblastic leukemia. Only two cases reported this mode of revelation but for lymphoma requiring a surgical intervention -as our case- firstly to relieve the patient by a radicular decompression and secondly for histological diagnosis.


Post-electric burn ileal perforation is a rare but severe complication leading to high morbidity and mortality if there is delay in diagnosis and management. We are describing a case of electric current injury of left forearm, chest, and abdominal wall with perforation of ileum in an 8-year old boy. Patient was successfully managed by primary closure of the ileal perforation. Electric injuries are of two types; low tension and high tension injuries with 1000 volt as a dividing line between them.[1] Electrical injury is produced by the conversion of electric energy into heat while passing through tissues.[2] Clinical manifestation can range from no apparent injuries to serious systemic damage. Abdominal visceral injuries are associated with high voltage injuries where the contact point is directly over the abdomen.[3] There can be direct consequences of the passage of electrical current through the abdominal viscera or a complication of electric injury such as curling ulcer. These intra-abdominal electric injuries are difficult to diagnose. Abdominal visceral injuries associated with low voltage current are a rare.[4]An 8-year old male child presented after 10 days of sustaining electric burn over left forearm, chest, and abdominal wall. He sustained electrical burn accidently after touching electrical cable which was described as low voltage current. After sustaining electric burn, patient was brought to the private hospital. They managed patient with dressing and medications. There were no abdominal complaints at that time so he was discharged. After a week he developed abdominal pain with distension. On examination, there was necrosis of skin with eschar on the left lower abdomen with entry wound on left forearm and exit wound over left lower abdomen. He was dehydrated with pulse 100/mint. It was 2nd degree burn. Abdomen was distended and tender. On investigation hemoglobin was 11.7 gm%, TLC- 17000/mm3, serum electrolytes, liver function test, serum amylase and lipase were in normal limits. In urine analysis albumin was present. ECG was normal. Ultrasound abdomen showed thick inter-loop collection, dilatation of small bowel loops, and collapsed colon. X-ray abdomen revealed free gas under both domes of diaphragm. He was diagnosed as having bowel perforation leading to peritonitis. Immediate exploratory laparotomy was performed. There was around 50 cc of bilious fluid in peritoneal cavity with plenty of flakes over intestine. On further exploration, a perforation (0.5x0.5cm) found in the proximal ileum. Refreshing of the margins of perforation was done and primary closure performed . Patient recovered uneventfully and was discharged on 7th postoperative day. Electric burn healed at the time of discharge. At follow up patient remained healthy.Electrical burns account for approximately 5% of the hospital admissions.[5] During electrical injury, current is passed through underlying structures and cause coagulative necrosis. During passage of current passes through tissues, heat is generated according to Joule’s law (heat = I2 current × R resistance). The increase in temperature causes the denaturation of the macro molecules which is usually irreversible.[6] The resistance of the tissues during the passage of an electrical current is variable (lower for nerves and vessels and higher for fat and bones). The trauma as a result of an electrical current varies according to the individual susceptibility and the quality of care provided at the site of the accident.[7]Few cases of entero-cutaneous fistulae and bowel perforation following electric current are reported in literature.. Abdominal visceral perforation seems to be a direct consequence of the electric current rather than a complication of the injury, and often associated with high voltage current. Abdominal visceral injury is rarely associated with low voltage current, perhaps because the current density and hence heat dissipated are comparatively less. In our case the injury was due to a low voltage current. The part of bowel injured was near to the exit wound. A very small area might have affected by the electric current which would have given way after a week as our patient remained asymptomatic for more than a week.In visceral injuries the colon and small intestine are the organs most frequently affected. Depending upon the extent of the perforation, anatomical site and presence of diffuse or localized peritonitis, the treatment vary from primary closure with or without protective colostomy to exteriorization and the Hartmann operation.[9] In our case there was minimal contamination so primary closure of ileum was done.The purpose of this report is to point out the potential for serious gastrointestinal damage associated with not only high voltage electrical injury but also with low voltage electrical current. Visceral injuries are rare in electrical burns, but they should be suspected when abdominal symptoms appear. A prompt diagnosis and timely intervention usually result in favourable outcome.Source of Support: NilConflict of Interest: None declared


Central odontogenic fibroma (COF) is an extremely rare benign tumor that accounts for 0.1% of all odontogenic tumors. It is a lesion associated with the crown of an unerupted tooth resembling dentigerous cyst. In this report, a 10-year-old male patient is presented, who was diagnosed with central odontogenic fibroma of simple type from clinical, radiological, and histopathological findings. The 1992 WHO classification defines a central odontogenic fibroma (COF) as “a fibroblastic neoplasm containing varying amounts of apparently inactive odontogenic epithelium” [Radiographically, COF usually appears as a unilocular radiolucency with well-defined borders but may also exhibit a multilocular appearance with scalloped margins . The tumA 10-year-old male patient presented with a painless swelling in the left side of the lower jaw of six-month duration which had gradually increased in size.Extra oral examination revealed facial asymmetry with a hard swelling on the left side of the lower jaw. Intraoral examination revealed swelling extending from distal aspect of the 36 to retromolar area. There was no evidence of paresthesia. On palpation, swelling was firm in consistency.Orthopantomogram (OPG) revealed a large well-defined unilocular radiolucent lesion surrounded by sclerotic border on left side of mandible extending anteriorly from developing 35 to half of the ramus posteriorly and superiorly close to coronoid process to inferiorly lower border of mandible. Developing 37 was pushed to the lower end of the radiolucent area .The differential diagnosis at this stage included dentigerous cyst, odontogenic keratocyst, and ameloblastoma. An incisional biopsy of the lesion was performed for the histopathological examination.Gross examination of the biopsy specimen showed several soft tissue bits measuring about 1 × 1 cm in size, whitish in color and firm in consistency.Microscopic examination revealed tumor composed of inactive appearing odontogenic epithelial islands in the form of long strands or isolated nests set in a background of myxoid to cellular connective tissue stroma. Connective tissue also showed fine fibrocollagenous matrix .Based on clinical, radiographic, and histopathological findings, a diagnosis of central odontogenic fibroma of simple type was established. The patient was then subjected to enucleation of the lesion and had been followed up for one year postoperatively and there was no recurrence noticed.COF is an extremely rare benign neoplasm that is most often found in females and the incidence between maxilla and mandible is 1 : 1 . It is fRadiographically it is mostly unilocular, but, however, larger lesions show multilocular radiolucency with well-defined borders, sometimes with radiopaque areas noted in the interior of the lesion.Multilocular lesions usually show more aggressive behavior with complications of resorption of teeth, radicular displacement of adjacent teeth, and expansion of cortical bones.Majority of COFs are clinically asymptomatic causing slow expansion of the cortical bones. Clinical signs often observed are prominence of vestibular cortical and lingual bone (75%), pain (50%), and rhizolysis  (37.5%) . Our casClinical differential diagnosis includes cysts of odontogenic origin, ameloblastoma, adenomatoid odontogenic tumor, and ameloblastic fibroma. The correct diagnosis is often arrived after histological examination of the lesion .According to the latest classification of odontogenic tumors reported by Gardner, the odontogenic fibroma is classified as a benign lesion derived from “odontogenic ectomesenchyme with or without odontogenic epithelium.” He also has referred the tumor made up of connective tissue and odontogenic islands resembling dental follicle as the simple type and to the tumor described by the WHO as the WHO-type COF .The current classification of odontogenic fibroma by WHO (2005) is (1) the WHO variant, and (2) the non-WHO variant. The WHO variant is considered as a mesenchymal odontogenic tumor and is comprised of two distinct cell types, a fibrous element, and an epithelial component that resembles dental lamina or its remnants. In contrast, the non-WHO variant lacks an epithelial component and is said to be a monomorphic fibroblastic tumor, purported to be of odontogenic mesenchymal origin and ostensibly derived from pulpal or follicular fibroblasts .Histologically the simple type is characterized by a tumor mass made up of mature collagen fibers interspersed usually by many plump fibroblasts that are very uniform in their placement and tend to be equidistant from each other. Small nests or islands of odontogenic epithelium that appear entirely inactive are present in variable but usually in quite minimal amounts. WHO type consists of relatively mature but quite cellular fibrous connective tissue with few to many islands of odontogenic epithelium. Osteoid, dysplastic dentin, or cementum-like material is also variably present. Our case resembled the Simple type.The histological differential diagnosis of COF includes ameloblastic fibroma (AF), desmoplastic fibroma (DF), and myxoma. AF is composed of epithelial component in the form of strands and islands, showing peripheral layer of cuboidal or columnar cells, which may enclose a small number of cells resembling stellate reticulum, and connective tissue component in AF is more cellular and embryonic looking. DF on the other hand shows aggressive behavior and is devoid of epithelial component and the fibroblasts are myofibroblastic in nature . The preThe treatment of COF is conservative surgery by the enucleation of the lesion. Recurrence is not common. Causes of recurrence are not related to histologic type but due to an incomplete surgical removal of the lesion .The purpose of presenting this case is to highlight the importance of histopathological examination of every tissue submitted for arriving at confirmatory diagnosis in addition to clinical and radiographic findings.


Intraosseous mucoepidermoid carcinoma is a rare tumor which affects women more than men and is more common in the mandible. The radiological examination reveals a well-defined unilocular or multilocular radiolucent lesion. This tumor may resemble a glandular odontogenic cyst, particularly in incisional biopsies. The accurate diagnosis of these lesions is imperative because the subsequent treatment of each lesion would be different. The purpose of this study is to report two cases of intraosseous mucoepidermoid carcinoma and explicate the differentiating criteria of this lesion from the glandular odontogenic cyst.  Intraosseous mucoepidermoid carcinomas (MEC) of the jaws are rare, establishing 2-3% of all testified mucoepidermoid carcinomas in the literature -2. This Intraosseous MEC affects females more than males and apparently implicates the mandible more than the maxilla . It may It has been reported in all age; it is more prevalent in 4th and 5th decades of life . Wide loGlandular odontogenic cyst (GOC), a rare lesion, constitutes 0.2% of all odontogenic cysts . This tuThe purpose of this report was to depict two cases of intraosseous mucoepidermoid carcinoma and elucidate the possible ways to differentiate this lesion from a glandular odontogenic cyst. A 44-year-old woman was referred to Jahad Clinic- Ahvaz, Iran, exhibiting a painless swelling in the left posterior region of the mandible endured for 2 years. The entire molar teeth were extracted in this region. The radiological examination illustrated a large, well-defined, multilocular radiolucent lesion with scalloped borders extending from the first molar area into the ascending ramus ; which pNo palpable lymph node was noticed in the neck on extraoral examination. A provisional diagnosis of odontogenic tumors was verified and subsequently an incisional biopsy was performed. The microscopic examination revealed islands of epidermoid, mucous and intermediate cells with cystic areas in a fibrous stroma . The finA 56-year-old edentulous woman was referred to Jahad clinic, Ahvaz, Iran,  complaining of a painless expansion of right posterior mandible endured for 11 months. Radiological assessment illustrated a large, well-defined multilocular radiolucent lesion, extending from the posterior body of the mandible into the ascending ramus . On extrThe criteria that acknowledges the diagnosis of intraosseous MEC is entailed as: (a) intact cortical plates while the cortical perforation does not exclude the diagnosis, (b) radiological evidence of bone destruction, (c) exclusion of an alternative primary tumor, which its metastasis could histologically resemble the central MEC, (d) exclusion of an odontogenic tumor and (e) histopathologic confirmation , 5. In tThe radiological features are diverse and presumably non-diagnostic, usually presented as a unilocular or multilocular radiolucency. Even though it is frequently scalloped; the margins of the lesion are often well defined . To the Chan et al.  stated tThe presence of calcifications was reported in the clear-cell variant and the conventional MEC, occurring in enduring neoplasms. The dystrophic calcification of the amorphous-eosinophilic material secreted by intermediate basal cells may perhaps produce these features -15. EverHistopathologically, GOC may be confused with intraosseous MEC -9. This Pires et al.  stated tThey compared CK expression in GOC and central MEC and noticed dissimilarities in CKs 18 (30% versus 100%) and 19 (100% versus 50%). Central MEC and GOC are perhaps distinct lesions with CK profiles comparable to lesions that have glandular and odontogenic origins, respectively. Moreover, expression of CKs 18 and 19 could be beneficial in their subsequent differential diagnosis .TORC1/MAML2 and MECT1:MAML2 gene fusion in intraosseous MEC was reported by studies of Khan et al.  and FowlThe foremost treatment for intraosseous MEC is wide local resection, enblock resection or hemi-mandibulectomy , 20. SelRadiotherapy seems to be a useful supplementary aid in cases represented with close surgical margins and high grade tumors . MicroscIn He et al.’s study , all patThe recurrence rate of this entity varies from 13-50% in different studies . MetastaIntraosseous MEC may resemble GOC; therefore, pathologists and surgeons should deliberate the diagnosis of central MEC in their mind once the pathology report signifies GOC. This is particularly imperative when the lesion is located in the posterior of the mandible. Although many case reports outlines the radiological features of this entity, the diagnosis should be established on clinical and pathologic characteristics and local or radical resective surgery would probably be the first option for patient treatment. Immunohistochemistry might be useful in the diagnosis of complicated cases or small specimens. Concerning the diagnosis of GOC, obtaining serial sections of the lesion is highly recommended.


Tuberculosis of the vulva is very rare. It is found in about 0.2% of the cases of genital tract tuberculosis. It usually presents as small shallow ulcers and multiple sinus tracts or rarely as elephantiasis of vulva. Except for very rare cases of primary tuberculosis in the vulva, it is usually associated with tuberculosis elsewhere in the body leading to secondary tuberculosis. Here, we report a case of secondary vulval tuberculosis which presented as a vulval mass in a 40-year-old female patient. The rarity of this presentation in the female genital tract is emphasized. Tuberculosis is a major health problem in India. Tuberculosis of genital tract constitutes about 0.2% to 2% of all gynecological cases. It is usually asymptomatic and mainly affects young women in the reproductive age group. Many authors opined that there may be hormone dependence of infection, as 80% of cases of tuberculosis of genital tract occur in the reproductive age group –3. It frA female patient aged 40 years old presented with complaints of vulval growth, multiple nodules with opening of sinuses, and yellowish white discharge since two years. Culture of discharge material from sinus was positive for staphylococcus bacilli. Based on these complaints and culture findings, patient was administered several courses of antibiotics. However, patient did not respond to the antibiotic treatment. Her past clinical history revealed that she had pulmonary tuberculosis since 15 years, for which she had taken an incomplete course of antitubercular treatment. She denied any premarital or extramarital sexual relationship. Her menstrual cycles were regular. Her obstetric history was normal with two living children. On examination, hypertrophied cauliflower like mass covering the entire labia majora was noted. Both sided inguinal lymph nodes were discrete and nontender. Ultrasonography of lower abdomen revealed no abnormal findings in bilateral fallopian tubes, ovaries, or uterus. Routine blood and biochemical investigations of patient were within normal limits. Partial vulvectomy procedure was done and the specimen was sent for histopathology study. Macroscopy. A skin covered tissue mass measuring 9 × 4 × 2.5 cm was received. Skin surface was nodular with openings of sinuses over some nodules. Cut surface was pale-white glistening and predominantly solid in appearance with tiny foci of capillary sized blood vessels filled with blood (Figures  Figures  and 2. Microscopy. Pseudoepitheliomatous hyperplasia of epidermis was noted in the microscopy. Dermis was loose edematous and showed fibrocollagenous and fibrovascular tissue with focal and diffuse mononuclear cell infiltration. Focal myxoid areas and few granulomas formed of epithelioid cells and Langhans giant cells surrounded by lymphocytes, plasma cells, and histiocytes were also noted (Figures  Figures  and 4. ZTubercular lesion of vulva usually presents as small shallow ulcers and multiple sinus tracts. Also it rarely presents as elephantiasis of vulva or hypertrophic vulval tuberculosis . In the Hematogenous or lymphatic dissemination from an active site of infection is the most common mode of spread of tubercular infection in female genital tract. Infection may also spread from contiguous intra-abdominal sites through fallopian tubes to female genital tract, or it can also be primary tuberculosis of vulva by direct inoculation of bacilli by a male partner suffering from tubercular lesion in epididymis and seminal vesicles. Tubercular vulvitis is usually associated with tuberculosis of upper genital tract. Except for very rare cases of primary inoculation in the vulva, vulval tuberculosis is usually associated with tuberculosis elsewhere in the body , 6. In tDetailed and thorough medical history recording, proper clinical examination, and presence of granulomatous reaction in microscopic examination can act as reliable diagnostic tools in detecting and managing vulval tuberculosis in patients presenting with unusual findings and help in prevention of patient undergoing unnecessary surgical interventions.


The patient was a 46-year-old man who had undergone resection for a bulky retroperitoneal tumor 16 years previously during a follow-up for von Recklinghausen’s disease. Histopathological examination of the resected specimen showed that the tumor was an extra-adrenal paraganglioma. After the surgery, he had survived without any recurrence of the tumor. However, 16 years after the initial surgery, liver tumors were identified, and he was referred to our hospital for further investigation and treatment. Abdominal imaging modalities showed three masses in the left lateral segment of the liver. Fluorodeoxyglucose-positron emission tomography/computed tomography showed an abnormal uptake of fluorodeoxyglucose corresponding to the mass lesions. The patient was diagnosed with a metastatic paraganglioma based on histopathological examination of a liver mass biopsy. The patient underwent left lateral sectionectomy of the liver. Histopathological examination of the resected specimen revealed proliferating cells with basophilic cytoplasm and oval densely stained nuclei arranged in an alveolar pattern, which was similar to the findings of the initial resection specimen. Immunohistochemical staining was positive for synaptophysin and chromogranin A. Based on these findings, the resected tumors were histopathologically diagnosed with liver metastases from the retroperitoneal paraganglioma. We concluded that this is an extremely rare case of liver metastases occurring long after the initial resection of extra-adrenal peritoneal paraganglioma with von Recklinghausen’s disease. Although the association of pheochromocytoma with von Recklinghausen’s disease is well recognized, the association of paraganglioma  with von Recklinghausen’s disease is rare , 2. To tThe patient was a 46-year-old man.None.At the age of 45, the patient was diagnosed with hypertension and cerebral aneurysm and was being followed up without treatment.Sixteen years ago (at the age of 30), the patient was found to have a bulky retroperitoneal paraganglioma measuring 20 cm during a follow-up of von Recklinghausen’s disease and underwent surgery in our department. The tumor, which had hemorrhagic necrosis, measured 25 × 17 × 15 cm and was located in the retroperitoneum  were within normal limits.Three space-occupying lesions with high echo level  were observed in the left lateral segment of the liver.Plain and enhanced computed tomography (CT) showed that three hypervascular masses, the largest of which was 2.9 cm in size, were located in the left lateral segment of the liver. Plain CT showed low-density areas. Contrast-enhanced CT showed early staining and washout in the portal-venous phase  showed that three masses with marginal enhancement were located in the left lateral segment of the liver, with no uptake of the contrast agent in the hepatocyte phase Fig. c, d.Abnormal uptake of fluorodeoxyglucose  was observed in the lesions identified by the above imaging studies. There was no significant abnormal FDG uptake in other regions.Liver mass biopsy was performed for a definite diagnosis. Proliferating cells with weak basophilic cytoplasm and oval densely stained nuclei arranged in an alveolar pattern, similar to the findings of the initial resection paraganglioma specimen, were observed. Immunohistochemical staining was positive for synaptophysin and chromogranin A and negative for HAS and cytokeratin AE1/AE3. Based on these findings, the patient was diagnosed with liver metastases of the extra-adrenal retroperitoneal paraganglioma.The patient underwent left lateral sectionectomy for the liver metastases from the paraganglioma. A laparotomy using an upper abdominal median incision plus high transverse incision was performed under general anesthesia. The adhesion from the previous surgery was separated. Three tumors were confirmed by palpation and intraoperative ultrasonography. There were no other metastatic lesions. The left lateral segment of the liver was resected. Macroscopic examination of the resected specimen showed whitish well-circumscribed solid tumors with capsules, with no invasion of the surrounding tissues Fig. .Fig. 3MaAlthough the Zellballen pattern typical of paraganglioma was not apparent, the tumors were composed of large and irregular nests of cells with anisokaryosis and basophilic cytoplasm Fig. a, which After the surgery, the patient had a favorable clinical course without complications. At the time of this report (6 months after the surgery), the patient has experienced no relapses and is being followed up on an outpatient basis.von Recklinghausen’s disease is an autosomal dominant disease first described by von Recklinghausen’s in 1882 . The majStudies have reported that distant metastasis is not uncommon in paragangliomas irrespective of the presence of von Recklinghausen’s disease. Sclafani et al.  reportedWe report a resected case of liver metastases from extra-adrenal retroperitoneal paraganglioma in a patient with von Recklinghausen’s disease 16 years after the initial surgery.Written informed consent was obtained from the patient for publication of this case report and any accompanying images.


Clostridium difficile was found to have portal venous gas but its significance was confounded by a recent surgery. Serial ultrasonographic exams triggered a surgical intervention. In the second case, we report what we call the “liver sign” a finding in patients with pneumoperitoneum. These findings, all obtained prior to conventional abdominal imaging, had immediate clinical impact and avoided unnecessary delays and radiation. Detection of abdominal air should be part of the routine-focused ultrasonographic exam and for critically ill patients an algorithm is proposed.In critical care patients, point of care abdominal ultrasound examination, although it has been practiced for over 30 years, is not as widespread as its cardiac or pulmonary counterparts. We report two cases in which detection of air during abdominal ultrasound allowed the early detection of life-threatening pathologies. In the first case, a patient with severe The online version of this article (doi:10.1186/s13089-016-0039-7) contains supplementary material, which is available to authorized users. In 2009, the American College of Chest Physicians (ACCP) and La Société de Réanimation de Langue Française (SRLF) proposed guidelines in critical care ultrasound . Those gClostridium difficile toxin assay returned positive. He was admitted to our intensive care unit (ICU) for monitoring and medical therapy with intravenous metronidazole and oral vancomycin. The patient improved from a hemodynamic, renal as well as abdominal pain point of view, but his WBC and creatinine started to increase 3 days later without new hemodynamic compromise. An abdominal ultrasound (US) was performed and revealed evidence of thumbprinting  was 80/40 mmHg and was tachycardic but displayed normal mentation. His examination was remarkable for diffuse abdominal pain and rebound tenderness, but no guarding. Laboratory work revealed acute kidney injury with a creatinine of 180 mmol/L. His pre-operative baseline value was 66 mmol/L. However, his arterial lactate and white blood cell counts (WBC) were normal. The patient received initial fluid resuscitation and underwent a computed tomography scan that showed a dilated cecum (8 cm) and an edematous colon. A The ICU team was called for immediate assessment of a 61-year-old woman who was hospitalized on the surgical ward with a complaint of desaturation and acute abdominal pain following dilation of a known anal stenosis which occurred 5 days prior. She was otherwise known for multiple abdominal surgeries in the past, ischemic cardiomyopathy with a 45 % ejection fraction, dyslipidemia and history of drug abuse. At the time, she was known to be taking pregabalin, hydromorphone, acetaminophen, aspirin, atorvastatin, and prophylactic subcutaneous heparin. Upon initial assessment, she was afebrile, tachypneic, and normotensive. Her heart rate was 110/min, BP normal but her oxygen saturation was 85 % on 50 % inspired oxygen. Initial examination was aimed at ruling out an acute coronary syndrome with subsequent congestive heart failure, but an electrocardiogram was normal and a focused transthoracic echocardiography (TTE) did not reveal new wall motion abnormalities or a worsening in the cardiac function. As part of our routine ultrasound examination, we looked at the inferior vena cava (IVC) –9. In orIn the above cases, we used the ultrasound to observe three abnormal air patterns that should alert the clinician to an intra-abdominal pathology, namely intraperitoneal air (case#2), portal venous gas (case#1), and intramural air (case#1).Intraperitoneal air can be seen using the liver as an acoustic window. Indeed, the liver offers a unique echogenic window. Although the hepatic flexure of the colon can sometimes be found interposed, there is generally no bowel that creates artifact in the right upper quadrant. Free air will accumulate anteriorly in the supine position and create an air artifact interfering with the normal liver texture Fig. . It is oTraditionally, other studies have described the main ultrasonographic finding of free intraperitoneal air as peritoneal stripe enhancement with associated reverberation artifacts , 12 and However, our cases highlight two important points that contrast with the existing literature. First, we did not document a peritoneal stripe enhancement, but the reverberation artifacts were present. Second, the phased array probe was used rather than the typical abdominal convex or linear probe. Several handheld echo devices do not have an abdominal probe. Rather, the phased array probe has become more prominently used as it allows reliable examination of the cardiac structures, which remains at the core of the assessment of the unstable patient. In addition, abdominal, renal, and cranial settings are commonly available when using the cardiac-phased array probe.C. difficile colitis, such as in our first case, complications such as worsening bowel wall edema, bowel distention and pneumatosis intestinalis can help guide the clinician towards surgical therapy prior to an obvious macrohemodynamic deterioration which would occur after perforation. Furthermore, artifacts that can mimick pneumoperitoneum have been described such as pseudo-pneumatosis intestinalis and pseudo-thickened gut [Caution has to be exercised in the interpretation of this finding as free air usually resolves rapidly post-laparotomy or laparoscopy but can rarely persist for weeks . In C. dened gut . InteresPortal air or hepatic portal venous gas can result from mesenteric ischemia or other conditions associated with alteration of the intestinal mucosa . Using sSeveral technical limitations preclude complete visualization of the gastrointestinal tract. Ultrasound itself is operator-dependent. In a small study of 31 patients in the emergency room with abdominal pain in which five patients were found to have pneumoperitoneum by both CT imaging and abdominal X-ray, the sensitivity and specificity of a hand-carried ultrasound were 80 and 81 %, respectively . Hence, However, a recent study showed that senior physicians with experience in abdominal US can rapidly learn to identify patterns suggestive of pneumoperitoneum with almost 90 % accuracy . The stuThe two cases we report offer additional insight into the usefulness of adding a focused abdominal exam to the already well-established focused TTE as proposed by the ACCP, SRLF, and other organizations , 21, 22.Written informed consent was obtained from the patient for publication of this case report and any accompanying images.


Epidermoid cysts are rarely detected malformations in the oral cavity. Their development sites are the sublingual, submaxillary, and submandibular spaces. In this paper, we report a three-month-old infant who was admitted to our hospital due upper lip swelling. Magnetic resonance imaging showed that she had a two-centimeter cystic lesion and fusion defects of orbicularis oris muscle. The cyst was surgically removed and histopathological diagnosis was “epidermoid cyst.” In recent literature, we could not find reports related to orbicularis oris muscle fusion defects because of epidermoid cyst. Dermoid or epidermal cysts arise from aberrant localization of epithelial component of ectodermal tissue in fetal life or aberrant inoculation of epithelial tissue after trauma or surgery . These cA 3-month-old infant was brought to our clinic with swelling in the upper lip, which was present at birth. The patient's parents had no remarkable disease in their medical history and the patient's mother suffered from no disease and she did not have any medication during her pregnancy. The infant was healthy with no history of trauma or surgery.When the infant cried and smiled, smiling deformity appeared. Intraoral examination revealed nearly 2 cm swelling located under the philtral columns in the gingiva-buccal mucosal area of the upper lip . There wWhen the patient was 4 months old, the lesion was dissected from surrounding tissues through a midline incision made on the oral mucosa of the upper lip after performing orotracheal intubation under general anesthesia, and sinus tract was completely excised via subepidermal excision . Fusion Excision biopsy and histopathological examination of the resected specimen showed a cyst lined by stratified squamous epithelium filled with lamellated keratin . No skinThe etiology of epidermal cysts and dermoid cysts is unclear. Clinical features of craniofacial dermoid and epidermoid cysts are presented in varied ways, including infection, asymptomatic puncti, or seizure secondary to intracranial invasion. Many theories have been suggested , 9. It hTo the best of our knowledge, there is no similar case in the literature. Although there are pediatric cases in the literature with epidermal cyst lesions in the sublingual region, gingiva, palate, and uvula, many of them had history of trauma or surgical intervention , 8, 10. In recent literature, we could find no reports related to orbicularis oris muscle fusion defects because of epidermoid cyst. Due to muscle fusion defect in our case, the best treatment timing for the patient was planned mainly based on the repair time of cleft lip deformity. Thus, lip muscle could be repaired at appropriate time in the same session with the excision of lesion.


Carcinosarcomas of the ureter are rare biphasic neoplasms, composed of both malignant epithelial (carcinomatous) and malignant mesenchymal (sarcomatous) components. Carcinosarcomas of the urinary tract are exceedingly rare. We report a unique case of a carcinosarcoma of the ureter with a chondrosarcoma and small cell tumor component arising in a 68-year-old male who presented with microscopic hematuria. CT intravenous pyelogram revealed right-sided hydroureter and hydronephrosis with thickening and narrowing of the right ureter. The patient underwent robot-assisted ureterectomy with bladder cuff excision and subsequent adjuvant chemotherapy. The patient is disease-free at 32 months after treatment. We provide a brief synoptic review of carcinosarcoma of the ureter and bladder with utilization of immunohistochemical (IHC) stains and potential diagnostic pitfalls. Carcinosarcomas of the ureter are rare malignant neoplasms with only around a dozen case reports within the literature –14. TheyA 68-year-old male presented with microscopic hematuria on routine physical exam, and further work-up revealed right-sided hydronephrosis and hydroureter with a transition zone in the right ureter. The patient was referred to our institution for consideration of right-sided robot-assisted ureteral excision. Evaluation with ureteroscopy found a mass in the right mid to distal ureter that was biopsied and read as high grade urothelial carcinoma. The patient subsequently underwent robotic-assisted right distal ureterectomy with bladder cuff excision. Macroscopically, there was no distinct mass but an ulcerated and hemorrhagic mucosa in the right ureter extending 7 cm in length with concurrent stricture. Microscopic examination of this specimen revealed a biphasic neoplasm composed of both malignant epithelial and mesenchymal components . The epiImmunohistochemical studies demonstrated that the epithelial components were immunoreactive with CAM 5.2, cytokeratin 7, and focally with cytokeratin 20. The small cell carcinoma component was positive for CD56, but other neuroendocrine markers including synaptophysin and chromogranin were negative. The high grade chondrosarcoma component was not stained. The morphologic and immunoprofile was consistent with a carcinosarcoma. The tumor was muscle invasive and the pathologic stage was pT2NX, according to the AJCC 7th edition. The patient was given the option of adjuvant therapy secondary to the high grade nature of the malignacy with muscle invasion and concurrent small cell carcinoma component. He received three cycles of cisplatin and gemcitabine chemotherapy. After 32 months of follow-up, the patient is alive and disease-free with negative CT and PET scans.Carcinosarcomas are aggressive neoplasms that can occur anywhere in the body but commonly occur within the uterus in women and the bladder in men. These malignant tumors most often occur in older patients, greater than 60 years of age, and are more frequent in men than women (M : F ratio of 4 : 1) . CarcinoCarcinosarcoma of the ureter is a rare malignant tumor with poor outcomes involving frequent recurrence within months and death from disease within two years , 14. It Aggressive surgical management is the treatment of choice in management of patients with carcinosarcoma of the ureter. Literature reports suggest that nephroureterectomy is the recommended procedure for surgically treating these lesions . This poSecondary to the rarity of these lesions, low number of patients to conduct meaningful prospective studies, and lack of long term follow-up, there is no clear consensus on radiochemotherapy regimens and impact on survival. The literature suggests that the prognosis is universally poor and that neoadjuvant or adjuvant chemotherapy and radiotherapy may not improve prognosis , 14. HowGiven the concern for residual microscopic disease and final pathology in our case, the decision was made to proceed with adjuvant chemotherapy. A combination of cisplatin and gemcitabine was used. The organ of disease origin and type of epithelial component in carcinosarcomas may be important factors in deciding upon which regimen to use. In addition, some confounding variables which could account for the apparent ineffectiveness of chemotherapy regimens include improper histological classification, focality of the tumor, size and depth of invasion, distance from or involvement of the renal pelvis, and/or genetic makeup of the tumor itself (inherently aggressive and unresponsive to treatment). Our patient is currently disease-free 32 months after treatment, suggesting a role for combined surgical and oncological treatment in the management of these patients.Recognition of this rare entity is important for urologists and pathologists alike to accurately diagnose these lesions and treat them aggressively. Though overall prognosis remains poor for the majority of patients, our case suggests multimodal therapy may provide an avenue for prolonged survival in carcinosarcomas of the ureter.


Unresectable cholangiocarcinoma is poorly responded to chemotherapy, especially for the case refractory to gemcitabine and cisplatin. Here, we tested whether high expression of thymidine phosphorylase (TP) can be a predictive biomarker for the indicator for gemcitabine and doxifluridine combination chemotherapy in the cholangiocarcinoma refractory to gemcitabine and cisplatin.Immunohistochemical staining for TP was performed with a biopsy specimen. We accepted the result as positive when more than 10% of cancer cells were stained with moderate intensity.Here, we report 2 cases of TP-positive cholangiocarcinoma well controlled with gemcitabine and doxifluridine combination chemotherapy, which had been refractory to the first line treatment with gemcitabine and cisplatin combination chemotherapy. In Korea, it has been reported to be as high as 2.6%.2 Although the etiology and carcinogenesis are not fully elucidated, chronic inflammation in the bile duct plays an important role in the development of cholangiocarcinoma. Congenital bile duct deformity, primary sclerosing cholangitis, intrahepatic stone, parasite infection, chronic typhoid carriers, and papillary changes of the bile duct are the risk factors for the development of cholangiocarcinoma.4 Operable cholangiocarcinoma can be cured by radical surgery, but 5-year survival rate ranges from 10% to 40%. In inoperable cases, the prognosis was dismal, and the overall median duration of survival is less than 6 months.6Cholangiocarcinoma is a tumor originating from bile duct epithelial cells. According to the autopsy results, the worldwide incidence is 0.01% to 0.5%, which indicates that cholangiocarcinoma is a relatively rare tumor.7 Nonetheless, even in patients who have responded to this treatment, the median survival rate is shorter than 12 months, and thus new treatment methods are required.8 For the case refractory to gemcitabine and cisplatin combination chemotherapy, few optional therapeutic trials are available.For the palliation of the patients with inoperable cholangiocarcinoma, the gemcitabine-based combination chemotherapy is widely used; gemcitabine and cisplatin combination chemotherapy is one of the most commonly used regimens.9 because various cancer tissues have higher levels of TP expression than adjacent non-neoplastic tissue.11 In addition, phase II trials showed that combination chemotherapy of gemcitabine and capecitabine (which is converted to 5-deoxy-5-fluorouridine in the liver and/or tumor tissue) had response rates ranging 30% to 48%,14 suggesting that they have synergistic effects. From these results, we assumed that gemcitabine and doxifluridine combination chemotherapy can be effective in cholangiocarcinoma even it is refractory to gemcitabine and cisplatin combination chemotherapy if the cholangiocarcinoma expressed TP highly in tumor cells.5-Fluorouracil (5-FU) is one of the most commonly used chemotherapeutic agents for gastrointestinal cancer. Doxifluridine (5-deoxy-5-fluorouridine), a prodrug of 5-FU, which is converted to 5-FU via thymidine phosphorylase (TP), generates 5-FU preferentially in cancer,Here, we report the results of 2 cases of TP-positive cholangiocarcinoma well controlled by gemcitabine and doxifluridine combination chemotherapy after they had been refractory to the first line treatment with gemcitabine and cisplatin combination chemotherapy. These results suggest that TP positivity might be a candidate for the predictive marker for the gemcitabine and doxifluridine combination chemotherapy.2 Day 1 and 8, cisplatin 60 mg/m2 Day 1, every 3 weeks), and transferred to our hospital. At the time of admission, vital signs were within normal ranges. In peripheral blood test, leukocyte was 4920/μL, hemoglobin was 10.0 g/dL, and platelet was 359,000/μL. In serum biochemistry test, they were also within normal ranges. At the time of admission, tumor marker tests were not performed.A 62-year-old female patient was admitted for discomfort in the upper abdominal area as the chief complaint. The patient visited at “S” hospital for the symptom and diagnosed as cholangiocarcinoma. Although laparoscopic resection was attempted, surgery could not be performed because of the presence of peritoneal seeding. Then she was treated with 1cycle of gemcitabine and cisplatin combination chemotherapy  for the response evaluation. The abdominal CT revealed that the size of tumor increased as compare to baseline CT findings. For the next therapeutic plan, we performed immunohistochemical staining for TP with a surgical biopsy specimen, and the result, as shown in Figure 2 Day 1 and 8, doxifluridine 1200 mg divided 3 times p.o. daily, every 3 weeks), and administered 4 cycles. The abdominal CT scan revealed that shrinkage of main mass indicating partial response , and then transferred back to our hospital due to disease progression after chemotherapy. On admission, vital signs were within normal ranges. In peripheral blood test, leukocyte was 4927/μL, hemoglobin was 9.1 g/dL, and platelet was reduced to 49,000/μL. In serum biochemistry test, they were within normal ranges except that alkaline phosphatase was mildly elevated to 174 IU/L. Abdominal CT scan revealed 12-cm sized huge mass invading portal vein  was initiated. For 11 months, the cancer lesion had been controlled as stable disease as shown in CT scan (Figure On admission, gemcitabine and doxifluridine combination chemotherapy (gemcitabine 1000 mg/m7 Up to now no combination chemotherapy has been proved to prolong the survival of the patients with cholangiocarcinoma. Hence, many clinical trials have been carried out to find a new therapy. In clinical practice, gemcitabine-based chemotherapy has been widely used. However, in the case of cholangiocarcinoma refractory to gemcitabine and cisplatin combination chemotherapy, few therapeutic trials are available. Here, confirming the overexpression of TP in cancer tissue, we used gemcitabine and doxifluridine combination chemotherapy for these refractory cases to gemcitabine and cisplatin combination chemotherapy. We found that this regimen was tolerable and effective even though the effects should need to be evaluated in prospective clinical trials. In phase II trials, it has been reported that combination chemotherapy of gemcitabine and capecitabine had some efficacy against cholangiocarcinoma and its toxicities was mild.14 Here, we performed immunohistochemical staining before starting chemotherapy and found that both the case was as strong positive immunoreactivity to TP as the TP expressions in gastric cancer.15 We used the same criteria as we used in gastric cancer in the previous report.15 The percentage of the positive cell of both case were more than 30%. The high level of TP expression in tumor tissue theoretically bring about tumor-selectively high concentration of 5-FU, which can result in good response to doxifluridine-based combination chemotherapy and minimal toxicities from this regimen. In addition, it has been reported that TP expression can be a predictive marker for oral 5-FU prodrug, which will be converted to 5-FU by TP.17 The treatment efficacy of doxifluridine was actually proven in gastric cancer, colon cancer, and other digestive tract cancers.11 These finding supports our therapeutic plan. In the 2 patients, time to progression for this combination chemotherapy was 9 and 11 months and the overall survival was 22 and 16 months, which appear to be longer than the average survival period of inoperable cholangiocarcinoma patients who had been refractory to the 1st-line combination chemotherapy.Inoperable advanced cholangiocarcinoma is poorly responded to chemotherapy, and the prognosis was dismal with the median survival ranging 6 to 12 months.Clinical studies showing that the overexpression of TP enhances the treatment efficacy of doxifluridine or capecitabine have not been reported in cholangiocarcinoma, yet. This is the first study suggesting that the gemcitabine and doxifluridine combination chemotherapy should be used as the second-line treatment for palliation in the cholangiocarcinoma showing positive expression for TP. The reason why we used doxifluridine instead of capecitabine is that doxifluridine is covered by medical insurance in Korea.It is unclear that gemcitabine and doxifluridine therapy can be applied for all the cholangiocarcinoma because not only the frequency of the TP expression but also the efficacy of positive TP expression as predictive marker in cholangiocarcinoma has not been investigated yet. We are under investigation on the frequency of TP expression in cholangiocarcinoma. Further prospective clinical trial is warranted for this regimen to determine whether TP positivity can be a predictive marker for the gemcitabine and doxifluridine combination chemotherapy in cholangiocarcinoma.


Only ten cases of neonatal congenital segmental dilatation (CSD) of the colon have been described so far. We present a full-term female newborn with trisomy 21, ventricular septal defect, and gross abdominal distension. Plain abdominal radiographs revealed a huge cystic lesion occupying the left hemiabdomen. Upon laparotomy on day 4 a CSD of the distal sigmoid and proximal rectum was confirmed and resected. The proximal colon was exteriorized and the distal part closed as a Hartmann pouch. Histology confirmed a huge segmental dilatation of the sigmoid without dysganglionosis or pseudodiverticula, but normal intestinal architecture. After correction of the ventricular septal defect a low rectal end-to-end anastomosis could be performed at an age of 5 months. The postoperative course was uneventful. CSD of the sigmoid colon is extremely “rare to meet” and a “challenge to treat” in the newborn period, but clinical awareness of this entity prompts pediatric surgical success. Congenital segmental dilatation (CSD) of the colon represents a rare congenital malformation most often diagnosed in children beyond the neonatal period. So far, only ten cases of neonatal colonic segmental dilatation have been described in the literature .The main characteristics of CSD of the colon include a single large dilatation of the colon of variable length with an abundant serosal blood vasculature but lacking teniae coli, haustrations, and appendices epiploicae . HoweverHerein, we report the findings and the operative management of a CSD of the colon in a neonate with trisomy 21 and ventricular septal defect.A full-term female newborn was spontaneously delivered with a birth weight of 2,600 g in a peripheral hospital. Additional findings were trisomy 21 combined with a ventricular septal defect. At day 1 the patient showed increasing abdominal distension. Clinically, the patient had a normal anal orifice and passed meconium within 14 h after birth.The radiological work-up revealed a huge gas filled cystic lesion located in the left hemiabdomen and a consecutive displacement of the intestine . The patUpon laparotomy on day 4 of life a grossly dilated pouch-like segment of the sigmoid colon became evident reaching well beyond the peritoneal space of Douglas . No highAfter correction of the ventricular septal defect by cardiac surgery closure of the colostomy was planned at an age of five months. Prior to surgery an anorectal manometry was performed showing inconspicuous results. Additionally, on rectal examinations relaxations were present. Therefore, Hirschsprung's disease was ruled out and a low rectal end-to-end anastomosis  could be performed. The postoperative course was uneventful. At the latest outpatient visit two months following the closure of the colostomy the patient was free of symptoms passing daily stool.In 1959, CSD of the colon was described by Swenson and Rathauser in a child for the first time . Since tTo confirm the diagnosis of CSD of the colon Brawner and Shafer have reported the following five criteria: (I) lack of radiographically demonstrable motility of the dilated segment, (II) normally appearing and functioning colon both proximal and distal to the dilated segment, (III) absence of teniae coli in the dilated segment, (IV) normal ganglion cells, and (V) hypertrophy of circular and longitudinal muscle layers in the dilated segment . These cOur patient fulfilled four out of the five Brawner criteria. While normal ganglion cells could be demonstrated histologically, no hypertrophy of the muscles layers was seen compare . This fiThe exact pathogenesis of CSD of the colon is still a matter of debate. Available theories include intrauterine vascular incidents, defective organogenesis, strangulation of the intestine in the umbilical ring, neurogenic causes, and defective muscular development . A wide The common symptoms of children with CSD of the colon include abdominal distention, chronic constipation, and episodes of diarrhea and are therefore often treated in the line of suspected Hirschsprung's disease . HirschsThe recommended treatment consists of resection of the affected segment with either end-to-end anastomosis or creation of a proximally located stoma , 9. In cIn contrast to congenital pouch colon (CPC), intestinal duplication, and Hirschsprung's disease congenital dilatation of the sigmoid colon is extremely “rare to meet” and a “challenge to treat” in the newborn period, but clinical awareness of this entity prompts pediatric surgical success.


This case presentation examines a rare clinical entity: colorectal adenocarcinoma (CRC) metastasis to the tongue. CRC is among the least common tumors to metastasize to the oral cavity. Objectives for this case report are to (1) maintain a high index of suspicion for oral cavity tumors representing metastatic disease, (2) consider appropriate surgical and adjunctive interventions, and (3) recognize the significance of identifying the primary tumor via immunohistochemical staining. We present a case of a 57-year-old male with a history of stage IV rectal adenocarcinoma metastatic to the lung who presented to our clinic with a painful mass of the right lateral tongue that he noticed one month before. MRI of the neck revealed a mass involving the anterior two-thirds of the right tongue with irregular margins and an ipsilateral enlarged right jugulodigastric lymph node. The patient underwent right partial glossectomy with primary reconstruction and right modified radical neck dissection. Pathology confirmed poorly differentiated adenocarcinoma consistent with a colorectal primary with lymphovascular and perineural invasion. The tumor was staged as T2N1, and the patient was referred for chemoradiation. In this report, we discuss the presentation, diagnosis, and treatment of this uncommon disease, with a thorough review of the world literature. Metastasis of primary tumors located elsewhere in the body to the oral and maxillofacial region is rare, representing only 1% of all oral cancers . In the A 57-year-old male with a history of stage IV rectal adenocarcinoma metastatic to the lung presented to our outpatient clinic with a painful mass on the right lateral tongue that he noticed one month before.Elective colonoscopy 2 years before revealed a 2 cm ulcerated polyp at the first rectal fold positive for invasive poorly differentiated primary tumor. Three weeks after his colonoscopy, the patient underwent transanal abdominal robotic total mesorectal excision (TME) with proctosigmoidoscopy. Lymphovascular and perineural invasion were identified. There was no evidence of metastasis at this time and the patient received radiation and chemotherapy.CT of the chest/abdomen/pelvis eight months after the TME revealed a 1.5 × 0.8 cm spiculated mass in the left lower lung. Pathology confirmed moderately differentiated adenocarcinoma consistent from a colorectal primary tumor. He subsequently underwent video-assisted thoracoscopic surgery (VATS) with left lobectomy. Chemotherapy was offered but it was deferred by the patient.The patient presented to our clinic 7 months later with a painful mass of the right lateral tongue. Review of systems was negative and the patient denied any weight loss or fatigue. The patient is a nonsmoker.Oral examination revealed fullness of the right tongue; floor of the mouth and buccal mucosa were normal to palpation. MRI of the neck revealed a mass involving the anterior two-thirds of the right tongue with irregular margins and ipsilateral right jugulodigastric lymphadenopathy . Punch bIHC staining suggested a colorectal primary. Direct laryngoscopy and esophagoscopy revealed a 3 cm mass along the anterior two-thirds of the right tongue which did not cross the midline. It extended to the undersurface of the tongue but did not occupy the floor of the mouth. The posterior tongue was unremarkable. PET scan revealed hypermetabolic activity in the tongue and neck consistent with metastatic disease Figures .The patient underwent a right partial glossectomy with primary reconstruction and right modified radical neck dissection. Pathology revealed a poorly differentiated adenocarcinoma consistent with a colorectal primary with lymphovascular and perineural invasion. The tumor measured as 3 cm and was staged as T2N1. The patient was referred back to the medical and radiation oncologist for adjunctive treatment. Unfortunately, the patient was unable to complete his chemoradiation therapy and passed away with disease despite all efforts.Metastasis of primary tumors located elsewhere in the body to the oral and maxillofacial region is rare, representing only 1% of all oral cancers [Oral cavity metastasis classically involves lesions of the mandible, with one study identifying bony involvement in 18/24 patients and soft tissue invasion in only 6/24 patients . A revieSeveral theories attempt to explain the mechanism of metastasis to the oral cavity. Batson postulated that the valveless vertebral venous plexus serves as a conduit for bypassing filtration through the lungs and the increase in intrathoracic pressure directs blood flow towards the head and neck region from the caval and azygos venous systems . In a caOral soft tissue metastasis typically manifests as highly vascularized and hemorrhagic exophytic lesions and may be confused for pyogenic granuloma, peripheral giant cell granuloma, or fibrous epulis . DiffereImmunohistochemical staining, in conjunction with histopathology, helps aid in definitively differentiating a primary intraoral malignancy from a metastatic lesion with a distant primary tumor. Positive staining for cytokeratin 20 (CK20) suggests a gastrointestinal primary tumor, as most CRCs are positive for CK20 . CK20 anMost patients presenting with oral metastasis have a distant primary tumor already diagnosed and treated. However, van der Waal et al. found that, in 8/24 of patients, the oral lesion was diagnosed before the primary tumor, and in 3 of these patients, identification of the primary tumor remained unknown despite further investigations . They alLocal treatment of mandibular metastasis is nearly always by radiotherapy, whereas soft tissue metastasis is usually approached surgically . A literDue to the relatively uncommon occurrence of intraoral malignant soft tissue tumors, clinicians should maintain an index of suspicion for a metastatic origin. Further research is needed to elucidate mechanisms of distant tumor metastasis to the oral cavity, which may in turn inspire new treatment approaches and potentially improve survival outcomes.


Gelastic seizure was first described by Trousseau in 1877 and comes from the Greek word gelos (laughs), as laughter is the main feature [ A hamartoma is a benign focal lesion of normal tissue growing in a disorganised manner anywhere in the body; it may alter the surrounding tissue or not show any symptoms. In this case, it emerged in the temporal region and provoked gelastic seizures (Greek: ‘Gelos’ or ‘laughter’), nervous breakdowns that were first described by Trousseau in 1877 [Remember that normal laughter is an emotional reactive behaviour and a motivated motor action that involves the limbic system, hypothalamus, the temporal cortex, and several regions of the brain stem .These gelastic nervous breakdowns are not a response to motivated actions and go unnoticed by patients who are classified as ‘poorly educated’ and even ‘crazy’. They occur most often in the temporal rather than the front lobe and have also been described as precocious puberty and developmental delay in a study conducted by Tassinari .It is most frequently diagnosed during childhood and the majority of patients will develop intractable epilepsy or other types of seizures, such as complex partial, generalised tonic–clonic seizures, in addition to important psychiatric comorbidity .The development of new image diagnostic methods, such as magnetic resonance imaging (MRI) with spectroscopy or diffusion tensor (DTI) and positron emission tomography (PET), has supplemented the traditional methods, such as computer tomography, to better target treatment decisions and improve the prognosis of these patients.In general, treatment with medication only does not have good results. Surgical treatment combined with drugs has better results , althougThe patient was a female, six years old, left-handed, with gelastic seizures, uncontrolled despite being treated with two antiepileptic drugs at high doses. A simple axial tomography was performed on her and a hypo-dense lesion that shapes the temporal internal table was observed, subsequent to this, a nuclear magnetic resonance was ordered for better characterising of a lesion in the second right temporal gyrus with a cystic appearance see .This lesion grew slowly (about 2 mm) in the last six months see ; becauseAlthough this was a single case, the following data pointed more towards a benign than malignant diagnosis: temporal diploe remodelation adjacent to the lesion, cystic appearance, absence of oedema, minimum peripheral enhancement with gadolinium, slow growth (2 mm in the last six months), displacement of association arcuate fibres, and lack of radiopharmaceutical uptake.In addition, it should be noted that the growth of the injury forced us to determine bilateral activation of motor and somato-sensory areas as well as the dominance of the language area that was left, a finding that allowed an adequate surgical resection without complications or permanent neurologic damage, especially in the case of an infant.Even though gelastic seizures have been found without a specific clinical pattern, laughter and joy are two clinical elements that involve different and complex mechanisms; therefore, the mere presence of this type of crisis compels us to consider the possibility of a temporal hamartoma or less frequently a frontal hamartoma, without being able to screen out a low-grade glioma that grows faster, has oedema, and more importantly, it is clear that due to the high sensitivity of the histopathological study, this method is definitive.Temporal hamartoma is a rare event that occurs more frequently in children, therefore we feel that the image method helped us to follow up the injury until measurement modification and determine the dominant hemisphere with functional MRI for surgery without permanent neuronal damage  and with


A 51-year-old female underwent vitrectomy surgery to remove a group of spherical subretinal tumors beneath the detached retina. Hematoxylin and eosin staining and immunohistochemical findings showed that the characteristics of the tumor were consistent with a subretinal heterotopic respiratory epithelium. This is the first report of a respiratory epithelial heterotopia located in the subretinal space. The choristoma, as a rare intraocular mass, is considered to be a congenital tumor that is not normally present in the area in situ. Although most ocular choristomas develop at the surface of the ocular, such as the conjunctiva, sclera, and orbit, the choristomas can also be found in intraocular structures, including the iris, ciliary body, retina, and optic nerve. Among them, the dermoid and epidermoid cysts are most common choristomatous cysts in the orbit.Here, we present a case of a 51-year-old woman with a respiratory epithelial heterotopia located in the subretinal space for the first time. It is very likely to be a new type of intraocular choristoma.A 51-year-old postmenopausal female presented with the chief complaint of an inferior visual field defect in the right eye and decreased central vision lasting for one day without any other concomitant symptoms. The patient had no significant past medical history, and claimed to have no antecedent ocular trauma. Nothing abnormal was found upon general physical examination. Her best corrected visual acuity (BCVA) was 20/200, and the intraocular pressure was 18 mmHg. Nothing abnormal was found in the anterior segment, and the vitreous was clear, without cells or pigments. Biocular indirect ophthalmoscopy revealed focal retinal detachment involving the area from the macula to the supratemporal vascular arcades, with subretinal depigmentation and pigmentation. A group of spherical subretinal tumors was noted beneath the detached retina. The tumors were semi-translucent, pale, smooth-faced, lobulated, and stationary. Neither proliferous tissue nor retinal breaks were found in the area of retinal detachment Fig. . The subThe initial diagnosis was exudative retinal detachment and a subretinal neoplasm in the right eye.The retina reattached spontaneously seven days after hospitalization. To our surprise, the retina detached again and the subretinal fluid recurred in the same area three days after treatment with antibiotic eye drops. Visual acuity and intraocular pressure did not change. Due to the repeated recurrence of retinal detachment and the unknown nature of the subretinal neoplasm, the patient underwent vitrectomy surgery with informed consent. The subretinal neoplasm was removed, and the retina was reattached with a silicone oil intraocular tamponade during the operation. During the surgery, the subretinal neoplasm was found to be spherical, lobulated and smooth-faced, and to be connected to the white fibrotic tissue under the tumor by a slender peduncle without any synechia to the retina Fig. . The tumThe excised mass was fixed in 10 % formalin and embedded in paraffin. Several 6-μm sections were cut from each paraffin block, two sections were stained with hematoxylin and eosin (H&E) and periodic acid Schiff-Alcian blue, and the others were stained with various antibodies (immunohistochemistry (IHC)). Immunohistochemical staining was performed using the streptavidin-peroxidase system  according to the manufacturer’s instructions. Commercially available, pre-diluted monoclonal antibodies against the following antigens were used: cytokeratin (CK), nerve tissue protein S100, glial fibrillary acidic protein (GFAP) . H&E staining revealed that the tumor was solid and covered with pseudostratified ciliated columnar epithelium containing scattered goblet cells, which were prominently shown by Alcian blue stain  showed a complete orbital bone and paranasal sinus. Therefore, an implanted cyst can be ruled out.In summary, we report a rare case of subretinal heterotopic respiratory epithelium, which is confirmed by H&E staining, Alcian Blue staining, and IHC staining for CK, GFAP and S100. This case is the first report of an intraocular respiratory epithelial heterotopia, and the most likely reason for the formation of this tumor is an intraocular choristoma.Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.